Functional Movement Symptoms: a casual examination of the ...
Functional Movement Disorders of the Face: A Historical Review and Case SeriesStone J1, Hoeritzauer I1, Tesolin L2, Carson A1,3 1 Centre for Clinical Brain Sciences, University of Edinburgh, Dept Clinical Neurosciences, Western General Hospital, Edinburgh 2 Clinica Neurologica, Ospedale di Cattinara, Trieste, Italia3 Department of Rehabilitation Medicine, NHS Lothian, Edinburgh, UKWord count: Abstract: 76 Main Text: 2263Running head: Functional facial movement disordersKeywords: Functional Neurological Disorders; Facial; Dystonia; Spasm; Psychogenic; Conversion Disorder; Functional Movement DisordersCorrespondence to: Dr Jon Stone, Dept Clinical Neurosciences, Western General Hospital, Edinburgh EH4 2XU. Email: Jon.Stone@ed.ac.uk Tel 0131 537 2911 Fax 0131 537 1132Author contributions and acknowledgements:JS is supported by an NRS Career Fellowship from NHS Scotland.IH is supported by an Association of British Neurology/Patrick Berthoud Clinical Research Training Fellowship.All authors report no disclosures and have read and followed the journal’s ethical publication guidelines.AbstractFunctional facial dystonia or spasm has, in recent years, been recognised as a relatively common form of functional movement disorder. We describe historical 'forgotten' neurological literature indicating that this was a well described phenomenon by the early part of the 20th century but subsequently faded from awareness. We add data from our own series of 41 patients with functional facial dystonia to explore the clinical features and associated comorbidities of patients with this movement disorder. INTRODUCTIONThe last decade has seen increasing awareness of the fact that functional movement disorder may affect the muscles of the face. An initial report on four patients by Tan and Jankovic in 2001ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "abstract" : "Facial spasms that distort facial expression are typically due to facial dystonia, tics, and hemifacial spasm (HFS). Psychogenic facial spasms, however, have not been well characterized. The authors sought to 1) determine prevalence of psychogenic facial spasm in patients referred for evaluation of HFS and 2) draw attention to clinical characteristics and potential diagnostic pitfalls. Among 210 consecutive patients referred for evaluation of HFS, 5 (2.4%) received diagnoses of psychogenic facial spasm. All patients were female; mean age was 34.6 years (range 26-45) and mean symptom duration 1.1 years (range 2 wk-2 yr). Onset was left-sided in 3 patients, and the lid was the initial site affected in 2 patients. This series of patients shows that facial spasms, although usually of neurovascular etiology, may be the initial or only manifestation of a psychogenic movement disorder, often associated with an underlying depression.", "author" : [ { "dropping-particle" : "", "family" : "Tan", "given" : "E K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Jankovic", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "The Journal of neuropsychiatry and clinical neurosciences", "id" : "ITEM-1", "issue" : "3", "issued" : { "date-parts" : [ [ "2001" ] ] }, "page" : "380-384", "title" : "Psychogenic hemifacial spasm.", "type" : "article-journal", "volume" : "13" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[1]", "plainTextFormattedCitation" : "[1]", "previouslyFormattedCitation" : "[1]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[1], was followed by scattered reportsADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Dengenhardt", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Zadikoff", "given" : "C", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Psychogenic Movement Disorders", "editor" : [ { "dropping-particle" : "", "family" : "Hallett", "given" : "M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Jankovic", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "AE", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Cloninger", "given" : "CR", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Yudofksy", "given" : "SC", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "2006" ] ] }, "page" : "341-3", "publisher" : "Lippincott Williams & Wilkins", "publisher-place" : "Philadelphia", "title" : "Psychogenic facial spasm (the smirk) presenting as hemifacial spasm", "type" : "chapter" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[2]", "plainTextFormattedCitation" : "[2]", "previouslyFormattedCitation" : "[2]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[2]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Carson", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of Neurology, Neurosurgery & Psychiatry", "id" : "ITEM-1", "issue" : "10", "issued" : { "date-parts" : [ [ "2010" ] ] }, "page" : "e8--e9", "publisher" : "BMJ Publishing Group Ltd", "title" : "020 Psychogenic/dissociative/functional facial symptoms\u2014a case report", "type" : "article-journal", "volume" : "81" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[3]", "plainTextFormattedCitation" : "[3]", "previouslyFormattedCitation" : "[3]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[3] prior to a seminal case series of 63 patients by Fasano et al in 2012ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.25190", "ISSN" : "1531-8257", "PMID" : "23033125", "abstract" : "The facial phenotype of psychogenic movement disorders has not been fully characterized. Seven tertiary-referral movement disorders centers using a standardized data collection on a computerized database performed a retrospective chart review of psychogenic movement disorders involving the face. Patients with organic forms of facial dystonia or any medical or neurological disorder known to affect facial muscles were excluded. Sixty-one patients fulfilled the inclusion criteria for psychogenic facial movement disorders (91.8% females; age: 37.0 \u00b1 11.3 years). Phasic or tonic muscular spasms resembling dystonia were documented in all patients most commonly involving the lips (60.7%), followed by eyelids (50.8%), perinasal region (16.4%), and forehead (9.8%). The most common pattern consisted of tonic, sustained, lateral, and/or downward protrusion of one side of the lower lip with ipsilateral jaw deviation (84.3%). Ipsi- or contralateral blepharospasm and excessive platysma contraction occurred in isolation or combined with fixed lip dystonia (60.7%). Spasms were reported as painful in 24.6% of cases. Symptom onset was abrupt in most cases (80.3%), with at least 1 precipitating psychological stress or trauma identified in 57.4%. Associated body regions involved included upper limbs (29.5%), neck (16.4%), lower limbs (16.4%), and trunk (4.9%). There were fluctuations in severity and spontaneous exacerbations and remissions (60%). Prevalent comorbidities included depression (38.0%) and tension headache (26.4%). Fixed jaw and/or lip deviation is a characteristic pattern of psychogenic facial movement disorders, occurring in isolation or in combination with other psychogenic movement disorders or other psychogenic features.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "Alfonso", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Valadas", "given" : "Anabela", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Prashanth", "given" : "L K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "Anthony E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Munhoz", "given" : "Renato P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Morgante", "given" : "Francesca", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Duker", "given" : "Andrew P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Girlanda", "given" : "Paolo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bentivoglio", "given" : "Anna Rita", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Espay", "given" : "Alberto J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2012", "10" ] ] }, "page" : "1544-51", "title" : "Psychogenic facial movement disorders: clinical features and associated conditions.", "type" : "article-journal", "volume" : "27" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[4]", "plainTextFormattedCitation" : "[4]", "previouslyFormattedCitation" : "[4]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[4]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/B978-0-12-801772-2.00031-X", "ISBN" : "9780128017722", "ISSN" : "0072-9752", "PMID" : "27719855", "abstract" : "Functional movement disorders (FMDs) affecting the eyelids, tongue, and other facial muscles are often underrecognized because their phenomenology has not been fully characterized. Nevertheless, these disorders are more common than previously thought. In this chapter we will discuss the phenomenology as well as the clinical and instrumental diagnosis of facial FMDs. Facial FMDs should be considered when a patient exhibits any combination of the following features: (1) fixed unilateral facial contractions, especially with lower lip, with or without ipsilateral jaw involvement, of maximal severity at onset; (2) inconsistent features such as changes in side and pattern during or between examination; (3) associated somatoform or nonphysiologic sensory or motor findings; (4) reduction or abolition of facial spasm with distraction; (5) response to suggestion or psychotherapy; (6) rapid onset and/or spontaneous remissions; and (7) normal neurologic examination. Supportive features are young age, female gender, and associated medical conditions such as depression, headaches, facial pain, fibromyalgia, or irritable-bowel syndrome. Finally, the differential diagnosis with the organic counterparts will be also addressed, particularly with respect to blepharospasm, oromandibular dystonia, and hemifacial spasm.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tinazzi", "given" : "M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Handbook of clinical neurology", "id" : "ITEM-1", "issue" : "1888", "issued" : { "date-parts" : [ [ "2017" ] ] }, "page" : "353-365", "title" : "Functional facial and tongue movement disorders.", "type" : "article-journal", "volume" : "139" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[5]", "plainTextFormattedCitation" : "[5]", "previouslyFormattedCitation" : "[5]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[5]. Subsequent small seriesADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/S1474-4422(15)00226-4", "ISSN" : "14744465", "PMID" : "26581970", "abstract" : "Functional (psychogenic) neurological symptoms are frequently encountered in neurological practice. Cranial movement disorders-affecting the eyes, face, jaw, tongue, or palate-are an under-recognised feature of patients with functional symptoms. They can present in isolation or in the context of other functional symptoms; in particular, for functional eye movements, positive clinical signs such as convergence spasms can be triggered by the clinical examination. Although the specialty of functional neurological disorders has expanded, appreciation of cranial functional movement disorders is still insufficient. Identification of the positive features of cranial functional movement disorders such as convergence and unilateral platysmal spasm might lend diagnostic weight to a suspected functional neurological disorder. Understanding of the differential diagnosis, which is broad and includes many organic causes (eg, stroke), is essential to make an early and accurate diagnosis to prevent complications and initiate appropriate management. Increased understanding of these disorders is also crucial to drive clinical trials and studies of individually tailored therapies.", "author" : [ { "dropping-particle" : "", "family" : "Kaski", "given" : "Diego", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bronstein", "given" : "Adolfo M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Edwards", "given" : "Mark J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Stone", "given" : "Jon", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "The Lancet Neurology", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2015" ] ] }, "page" : "1196-1205", "title" : "Cranial functional (psychogenic) movement disorders", "type" : "article", "volume" : "14" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[6]", "plainTextFormattedCitation" : "[6]", "previouslyFormattedCitation" : "[6]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[6] have all added to the characterisation of functional facial movement disorders which were defined by Fasano and Tinazzi as a dystonia with fixed unilateral facial contraction, usually involving the lower lip and often with ipsilateral orbicularis oculis and jaw involvement. They are often of maximal severity at onset and display inconsistencies on clinical examination, such as resolution with distraction and changes in side and pattern during or between examination or spontaneous remissionsADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/B978-0-12-801772-2.00031-X", "ISBN" : "9780128017722", "ISSN" : "0072-9752", "PMID" : "27719855", "abstract" : "Functional movement disorders (FMDs) affecting the eyelids, tongue, and other facial muscles are often underrecognized because their phenomenology has not been fully characterized. Nevertheless, these disorders are more common than previously thought. In this chapter we will discuss the phenomenology as well as the clinical and instrumental diagnosis of facial FMDs. Facial FMDs should be considered when a patient exhibits any combination of the following features: (1) fixed unilateral facial contractions, especially with lower lip, with or without ipsilateral jaw involvement, of maximal severity at onset; (2) inconsistent features such as changes in side and pattern during or between examination; (3) associated somatoform or nonphysiologic sensory or motor findings; (4) reduction or abolition of facial spasm with distraction; (5) response to suggestion or psychotherapy; (6) rapid onset and/or spontaneous remissions; and (7) normal neurologic examination. Supportive features are young age, female gender, and associated medical conditions such as depression, headaches, facial pain, fibromyalgia, or irritable-bowel syndrome. Finally, the differential diagnosis with the organic counterparts will be also addressed, particularly with respect to blepharospasm, oromandibular dystonia, and hemifacial spasm.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tinazzi", "given" : "M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Handbook of clinical neurology", "id" : "ITEM-1", "issue" : "1888", "issued" : { "date-parts" : [ [ "2017" ] ] }, "page" : "353-365", "title" : "Functional facial and tongue movement disorders.", "type" : "article-journal", "volume" : "139" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[5]", "plainTextFormattedCitation" : "[5]", "previouslyFormattedCitation" : "[5]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[5]. The history of the field has not been one of linear accumulating knowledge. ‘Hysteria’ was a core part of neurological textbooks in the 19th century but it gradually lost its popularity as a subject of neurological study over the course of the 20th century ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1258/jrsm.2007.070129", "ISSN" : "0141-0768", "PMID" : "18263908", "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "Jon", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Hewett", "given" : "Russell", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Carson", "given" : "Alan", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Warlow", "given" : "Charles", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Sharpe", "given" : "Michael", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of the Royal Society of Medicine", "id" : "ITEM-1", "issue" : "1", "issued" : { "date-parts" : [ [ "2008", "1" ] ] }, "language" : "eng PT - Historical Article PT - Journal Article SB - IM", "note" : "DA - 20080211", "page" : "12-8", "publisher-place" : "Department of Clinical Neurosciences, University of Edinburgh, Western General Hospital, Crewe Rd, Edinburgh EH4 2XU, UK. jon.stone@ed.ac.uk", "title" : "The 'disappearance' of hysteria: historical mystery or illusion?", "type" : "article-journal", "volume" : "101" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[7]", "plainTextFormattedCitation" : "[7]", "previouslyFormattedCitation" : "[7]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[7]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/B978-0-12-801772-2.00003-5", "ISBN" : "9780128017722", "ISSN" : "00729752", "PMID" : "27719844", "abstract" : "The history of functional neurologic disorders in the 20th century from the point of view of the neurologist is U-shaped. A flurry of interest between the 1880s and early 1920s gave way to lack of interest, skepticism, and concern about misdiagnosis. This was mirrored by increasing professional and geographic divisions between neurology and psychiatry after the First World War. In the 1990s the advent of imaging and other technology highlighted the positive nature of a functional diagnosis. Having been closer in the early 20th century but later more separate, these disorders are now once again the subject of academic and clinical interest, although arguably still very much on the fringes of neurology and neuropsychiatry. Revisiting older material provides a rich source of ideas and data for today's clinical researcher, but also offers cautionary tales of theories and treatments that led to stagnation rather than advancement of the field. Patterns of treatment do have a habit of repeating themselves, for example, the current enthusiasm for transcranial magnetic stimulation compared to the excitement about electrotherapy in the 19th century. For these reasons, an understanding of the history of functional disorders in neurology is arguably more important than it is for other areas of neurologic practice.", "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "J.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Handbook of Clinical Neurology", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "2017" ] ] }, "page" : "25-36", "title" : "Chapter 3 \u2013 Neurologic approaches to hysteria, psychogenic and functional disorders from the late 19th century onwards", "type" : "chapter", "volume" : "139" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[8]", "plainTextFormattedCitation" : "[8]", "previouslyFormattedCitation" : "[8]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[8]. Consequently, a considerable number of useful but older clinical descriptions have been forgotten. Much of our apparently new knowledge in this field revisits clinical experience that had been documented in the past. Fasano et al noted that patients diagnosed with atypical facial movement disorders in studies going back to 1986 also probably fulfilled the diagnostic criteria for functional movement disordersADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.25190", "ISSN" : "1531-8257", "PMID" : "23033125", "abstract" : "The facial phenotype of psychogenic movement disorders has not been fully characterized. Seven tertiary-referral movement disorders centers using a standardized data collection on a computerized database performed a retrospective chart review of psychogenic movement disorders involving the face. Patients with organic forms of facial dystonia or any medical or neurological disorder known to affect facial muscles were excluded. Sixty-one patients fulfilled the inclusion criteria for psychogenic facial movement disorders (91.8% females; age: 37.0 \u00b1 11.3 years). Phasic or tonic muscular spasms resembling dystonia were documented in all patients most commonly involving the lips (60.7%), followed by eyelids (50.8%), perinasal region (16.4%), and forehead (9.8%). The most common pattern consisted of tonic, sustained, lateral, and/or downward protrusion of one side of the lower lip with ipsilateral jaw deviation (84.3%). Ipsi- or contralateral blepharospasm and excessive platysma contraction occurred in isolation or combined with fixed lip dystonia (60.7%). Spasms were reported as painful in 24.6% of cases. Symptom onset was abrupt in most cases (80.3%), with at least 1 precipitating psychological stress or trauma identified in 57.4%. Associated body regions involved included upper limbs (29.5%), neck (16.4%), lower limbs (16.4%), and trunk (4.9%). There were fluctuations in severity and spontaneous exacerbations and remissions (60%). Prevalent comorbidities included depression (38.0%) and tension headache (26.4%). Fixed jaw and/or lip deviation is a characteristic pattern of psychogenic facial movement disorders, occurring in isolation or in combination with other psychogenic movement disorders or other psychogenic features.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "Alfonso", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Valadas", "given" : "Anabela", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Prashanth", "given" : "L K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "Anthony E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Munhoz", "given" : "Renato P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Morgante", "given" : "Francesca", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Duker", "given" : "Andrew P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Girlanda", "given" : "Paolo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bentivoglio", "given" : "Anna Rita", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Espay", "given" : "Alberto J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2012", "10" ] ] }, "page" : "1544-51", "title" : "Psychogenic facial movement disorders: clinical features and associated conditions.", "type" : "article-journal", "volume" : "27" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[4]", "plainTextFormattedCitation" : "[4]", "previouslyFormattedCitation" : "[4]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[4]. From our own reading of the older literature, however, it was clear that this clinical entity had already been recognised much further back in the 19th century. In this article, we re-examine the historical literature on functional facial movement disorders and compare it, and recent work, with a new case series of 41 patients with functional facial movement disorders to extend the historical and clinical perspective of this clinical presentation.METHODSFor the historical review, we carried out a systematic search of a collection of neurological textbooks and books on hysteria and allied conditions published prior to 1920. All book titles are available for public download from . Each book was searched for the terms ‘facial’, ‘blepharospasm’ and ‘ocular’. In this section, we sometimes use the term hysterical when it was the term used in these publications. In addition, we searched for descriptions of patients with atypical facial dystonia or movement disorder from 1960 until 2017 who had features in keeping a functional facial movement disorder. For both the historical literature and recent search we explored references when relevant. Our case series is derived from 41 patients with functional facial movement disorders seen consecutively in general neurology and specialist ‘functional’ clinics in the Department of Clinical Neurosciences, Edinburgh by one of the authors (JS) over a period from 2008-2013. REVIEW OF LITERATURE 1880s to 1960sWe cannot find an earlier reference than 1887 when Charcot described unilateral hysterical facial spasm in the well-known patient 'Le Log' as followsADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Charcot", "given" : "J M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issue" : "3", "issued" : { "date-parts" : [ [ "1889" ] ] }, "number-of-pages" : "14-18", "publisher" : "New Sydenham Society", "publisher-place" : "London", "title" : "Clinical lectures on diseases of the nervous system (Volume 3)", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[9]", "plainTextFormattedCitation" : "[9]", "previouslyFormattedCitation" : "[9]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[9]: “...left labial commissure is raised and mouth is partly open. At first thought to be paralysis of right inferior facial...on further examination it is due to spasm of the muscles on the left side of the face”. The following year Charcot launched his new journal, Nouvelle Iconographie de Salpêtrière, which incorporated the latest photographic techniques. In the first issue is an article by Charcot and Paul Richer with drawings demonstrating this phenomenon, as well as showing tongue deviation to the affected side. The articles main purpose was to point out the similarity of the appearances to various types of mascaron, similar to a gargoyle on a church and used to frighten away evil spirits (Figure 1)ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Charcot", "given" : "J-M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Richer", "given" : "P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Nouvelle Iconographie de Salp\u00eatri\u00e8re", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1988" ] ] }, "page" : "87-92", "title" : "LE MASCARON GROTESQUE DE L'\u00c9GLISE SANTA MARIA FORMOSA, A VENISE ET L'H\u00c9MISPASME GLOSSO-LABI\u00c9 HYST\u00c9RIQUE", "type" : "article-journal", "volume" : "1" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[10]", "plainTextFormattedCitation" : "[10]", "previouslyFormattedCitation" : "[10]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[10]. The next volume of the journal in 1889 carried a long article by Tourette which included a description of “blépharospasm hystérique” with photographs of a patient with a typical ‘depressed’ eyebrow on the affected side, and showing subsequent recovery with the aid of hypnosis (Figure 1)ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Tourette", "given" : "G", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Nouvelle Iconographie de Salp\u00eatri\u00e8re", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1889" ] ] }, "page" : "107-120", "title" : "DE LA SUPERPOSITION DES TROUBLES DE LA SENSIBILIT\u00c9 ET DES SPASMES DE LA FACE ET DU COU CHEZ LES HYST\u00c9RIQUES", "type" : "article-journal", "volume" : "2" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[11]", "plainTextFormattedCitation" : "[11]", "previouslyFormattedCitation" : "[11]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[11]. In 1891 another article from Odessa carried another photograph of a patient with ‘hysterical’ hemifacial spasm (Figure 1).Gowers at around the same timeADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Gowers", "given" : "W R", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "A Manual of diseases of the Nervous System", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1892" ] ] }, "page" : "903-960", "publisher" : "Churchill", "publisher-place" : "London", "title" : "Hysteria", "type" : "chapter" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[12]", "plainTextFormattedCitation" : "[12]", "previouslyFormattedCitation" : "[12]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[12] also described hysteria affecting the face:"In hysteria there is either tonic contracture, especially in the orbicularis, or attacks of quivering movement, which do not resemble true facial spasm.... The effect of the preponderant contraction in the orbicularis and zygomatic muscles is a curiously mixed emotional aspect, a sort of whimpering smile ". Gowers, whose chapter on hysteria in that book has rarely been surpassed, also commented that these spasms were " usually lessened by rest, physical and mental... always increased by emotion, and by movement of the face, whether in speaking or chewing and ...by light and by cold." He noted that "The influence of light is intelligible, since the orbicularis palpebrarum is almost always involved, and a strong light produces reflex contraction in this muscle under normal circumstances". Gowers also refer to the presence of 'wrong way' tongue deviation, in which the tongue deviates to the side of facial spasm/apparent weakness, and is the opposite of what would be expected were the patient to have a pontomedullary lesion.Figure 1. Drawings and photographs from Nouvelle Iconographie de Salpêtrière, the in-house neurology journal of the Salpêtrière, founded by Jean-Martin Charcot. Upper images are from Volume 1 and show two patients with ‘l’hémispasme glosso-labié hystérique’ compared to a Venetian ‘mascaron’ ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Charcot", "given" : "J-M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Richer", "given" : "P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Nouvelle Iconographie de Salp\u00eatri\u00e8re", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1988" ] ] }, "page" : "87-92", "title" : "LE MASCARON GROTESQUE DE L'\u00c9GLISE SANTA MARIA FORMOSA, A VENISE ET L'H\u00c9MISPASME GLOSSO-LABI\u00c9 HYST\u00c9RIQUE", "type" : "article-journal", "volume" : "1" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[10]", "plainTextFormattedCitation" : "[10]", "previouslyFormattedCitation" : "[10]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[10]. Lower left and centre is a patient of Tourette with unilateral spasm treated with hypnosisADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Tourette", "given" : "G", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Nouvelle Iconographie de Salp\u00eatri\u00e8re", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1889" ] ] }, "page" : "107-120", "title" : "DE LA SUPERPOSITION DES TROUBLES DE LA SENSIBILIT\u00c9 ET DES SPASMES DE LA FACE ET DU COU CHEZ LES HYST\u00c9RIQUES", "type" : "article-journal", "volume" : "2" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[11]", "plainTextFormattedCitation" : "[11]", "previouslyFormattedCitation" : "[11]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[11] and lower right a patient of Bogroff in OdessaADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Bogroff", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Nouvelle Iconographie de Salp\u00eatri\u00e8re", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1890" ] ] }, "page" : "428", "title" : "CONTRIBUTION A L'\u00c9TUDE DES SYNDROMES HYST\u00c9RIQUES \u00ab SIMULATEURS \u00bb", "type" : "article-journal", "volume" : "4" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[13]", "plainTextFormattedCitation" : "[13]", "previouslyFormattedCitation" : "[13]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[13].Babinski and Froment summarise earlier descriptions under the term 'glosso-labial hemispasm’, under which it can often be found in subsequent textbooksADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Babinski", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Froment", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1918" ] ] }, "publisher" : "Univ of London Press", "publisher-place" : "London", "title" : "Hysteria or Pithiatism (trans. by JD Rolleston)", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[14]", "plainTextFormattedCitation" : "[14]", "previouslyFormattedCitation" : "[14]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[14]: “In glosso-labial hemispasm, described by Charcot, Brissaud and P. Marie, the spasm, as its name indicates, may be limited to the tongue and lips but sometimes affects simultaneously the orbicularis palpebrarum, platysma and neck muscles. The hook-like appearance of the tongue and the intermittent spasms of the contracted muscles give it an almost pathognomonic appearance.” They also reinforce the potential for confusion that could arise regarding whether there was paralysis or not “when there is facial asymmetry it will be found to be due not to muscular hypotonus but to spasm.”Dejerine commented 'as far as the face is concerned one much more frequently observes a glossolabial spasm than a facial paralysis properly so calledADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Dejerine", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Gauckler", "given" : "E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "edition" : "I", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1913" ] ] }, "number-of-pages" : "46-51", "publisher" : "J B Lippincott Co", "publisher-place" : "Philadelphia & London", "title" : "The Psychoneuroses and their treatment by psychotherapy", "type" : "book" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[15]", "plainTextFormattedCitation" : "[15]", "previouslyFormattedCitation" : "[15]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[15]’. Charcot and Dercum were not convinced that facial muscular paralysis could occur as a hysterical symptom stating ‘in the hysteric the deviation of the mouth and tongue, and facial paralysis, are wanting.’ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Charcot", "given" : "Jean Martin", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "The New Sydenham Society", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1877" ] ] }, "number-of-pages" : "Plate V, Vol I", "publisher" : "J.E. Adlard", "publisher-place" : "London", "title" : "Lectures on the disease of the nervous system", "type" : "book", "volume" : "XC" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[16]", "plainTextFormattedCitation" : "[16]", "previouslyFormattedCitation" : "[16]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[16] Preston in 1897,considered facial spasm to be 'not rare' but hysterical facial weakness 'very infrequent'ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Preston", "given" : "GJ", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1897" ] ] }, "publisher" : "P.Blakiston, Son & Co", "publisher-place" : "Philadelphia", "title" : "Hysteria and certain allied conditions", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[17]", "plainTextFormattedCitation" : "[17]", "previouslyFormattedCitation" : "[17]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[17]. Wood, in JAMA in 1898 ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Wood", "given" : "CA", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of the American Medical Association", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1898" ] ] }, "page" : "1136-8", "title" : "The methods employed in examining the eyes for the detection of hysteria", "type" : "article-journal", "volume" : "31" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[18]", "plainTextFormattedCitation" : "[18]", "previouslyFormattedCitation" : "[18]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[18]writing about neuro-ophthalmological aspects of hysteria commented, “a very common and in my opinion characteristic eye-sign in hysteria is spasm of orbicularis, the so-called blepharospasm…When this is unilateral it is almost invariably hysteric.” Pershing discusses this diagnosis in 1901ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Pershing", "given" : "HT", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1901" ] ] }, "publisher" : "P.Blakiston, Son & Co", "publisher-place" : "Philadelphia", "title" : "The diagnosis of nervous and mental diseases", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[19]", "plainTextFormattedCitation" : "[19]", "previouslyFormattedCitation" : "[19]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[19] and Oppenheim said he had seen it only once in 1900ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Oppenheim", "given" : "H", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Mayer (transl.)", "given" : "E E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1900" ] ] }, "publisher" : "J.B.Lippincott Co.", "publisher-place" : "Philadelphia & London", "title" : "Diseases of the Nervous System: A textbook for student and practitioner of medicine", "type" : "book", "volume" : "1st" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[20]", "plainTextFormattedCitation" : "[20]", "previouslyFormattedCitation" : "[20]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[20]. Janet, wrote that he had seen ‘many cases of hysterical facial paralysis’ that were ‘typical’ ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Janet", "given" : "P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1907" ] ] }, "publisher" : "Macmillan", "publisher-place" : "London", "title" : "The major symptoms of hysteria", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[21]", "plainTextFormattedCitation" : "[21]", "previouslyFormattedCitation" : "[21]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[21] but he may have been describing spasm since he doesn’t refer specifically to weakness. In the 1920 edition of Diagnosis of Nervous Diseases by Purves-Stewart there are three photographs of patients displaying functional unilateral blepharospasm (Fig.2)ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Purves-Stewart", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1920" ] ] }, "publisher" : "Arnold", "publisher-place" : "London", "title" : "The Diangosis of Nervous Diseases", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[22]", "plainTextFormattedCitation" : "[22]", "previouslyFormattedCitation" : "[22]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[22]. Figure 2: Functional facial movement disorder – historical cases. A-DADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Hurst", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1920" ] ] }, "publisher" : "Henry Frowde, Hodder & Stoughton, Oxford University Press", "publisher-place" : "London", "title" : "The psychology of the special senses and their functional disorders", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[23]", "plainTextFormattedCitation" : "[23]", "previouslyFormattedCitation" : "[23]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[23]: (A) Bilateral ptosis with frontalis overactivity, cured within several weeks. (B) Right ptosis requiring frontalis overactivity to see. Left orbicularis overactivity with amblyopia, resolved with a one-hour treatment (right hand image). (C) Right orbicularis overactivity and left frontalis overactivity with ptosis and amblyopia of five months duration, and after a half-hour treatment (left hand image). (D) Bilateral ptosis at rest with right facial spasm and apparent left facial weakness; left eyelid raised to see; attempt to open eyes resulting in overactivity of left frontalis and spasm of the right side of the face. (E) Functional facial spasm and torticollisADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Mott", "given" : "F W", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1919" ] ] }, "publisher" : "Henry Frowde, Hodder & Stoughton, Oxford University Press", "publisher-place" : "London", "title" : "War Neuroses and Shell Shock", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[24]", "plainTextFormattedCitation" : "[24]", "previouslyFormattedCitation" : "[24]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[24]. (F & G): Left ‘hemiglossolabial spasm’ associated with ipsilateral functional limb weakness and contractureADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Purves-Stewart", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1920" ] ] }, "publisher" : "Arnold", "publisher-place" : "London", "title" : "The Diangosis of Nervous Diseases", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[22]", "plainTextFormattedCitation" : "[22]", "previouslyFormattedCitation" : "[22]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[22]. Also at that time Arthur Hurst, a British physician known best for his films of patients with shell shockADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1093/jhmas/jrr015", "ISBN" : "00225045", "ISSN" : "1468-4373", "PMID" : "21596724", "abstract" : "From 1917 to 1918, Major Arthur Hurst filmed shell-shocked patients home from the war in France. Funded by the Medical Research Committee, and using Path\u00e9 cameramen, he recorded soldiers who suffered from intractable movement disorders as they underwent treatment at the Royal Victoria Hospital in Netley and undertook programs of occupational therapy at Seale Hayne in Devon. As one of the earliest UK medical films, Hurst's efforts may have drawn inspiration from the official documentary of the Battle of the Somme and films made in 1916 by French Army neurologists. Although initially motivated to make use of a novel medium to illustrate lectures, Hurst was alert to the wider appeal of the motion picture and saw an opportunity to position himself in the postwar medical hierarchy. Some \"before treatment\" shots were reenacted for the camera. Hurst, like some other shell shock doctors, openly used deception as a therapeutic measure. On the basis that the ends justified the means, they defended this procedure as ethical. Clinicians also took advantage of changes in military regulations to address functional symptoms. Claims made of \"cures\" in the film and associated publications by Hurst were challenged by other doctors treating shell shock. The absence of follow-up data and evidence from war pension files suggested that Hurst may have overstated the effectiveness of his methods. Nevertheless, the message conveyed in the film that chronic cases could be treated in a single session had a powerful resonance for ambitious or charismatic doctors and was revived in World War II.", "author" : [ { "dropping-particle" : "", "family" : "Jones", "given" : "Edgar", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of the history of medicine and allied sciences", "id" : "ITEM-1", "issue" : "3", "issued" : { "date-parts" : [ [ "2012", "7" ] ] }, "page" : "345-73", "title" : "War neuroses and Arthur Hurst: a pioneering medical film about the treatment of psychiatric battle casualties.", "type" : "article-journal", "volume" : "67" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[25]", "plainTextFormattedCitation" : "[25]", "previouslyFormattedCitation" : "[25]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[25]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "23538744", "ISSN" : "2160-8288", "PMID" : "23538744", "abstract" : "BACKGROUND The psychological pressure on soldiers during World War I (WWI) and other military conflicts has resulted in many reported cases of psychogenic gait as well as other movement disorders. In this paper, psychogenic movement disorders captured in the WWI film footage \"War Neuroses\" is reanalyzed. METHODS Two movement disorders specialists re-examined film images of 21 WWI patients with various and presumed psychogenic manifestations, pre- and post treatment. The film was recorded by Arthur Hurst, a general physician with an interest in neurology. RESULTS All 21 subjects were males, and all presented with symptoms relating to war trauma or a psychological stressor (e.g., being buried, shrapnel wounds, concussion, or trench fever). The most common presenting feature was a gait disorder, either pure or mixed with another movement disorder (15), followed by retrograde amnesia (2), abnormal postures (pure dystonia) (1), facial spasm (1), head tremor (1), \"hyperthyroidism-hyperadrenalism\" (1). Nineteen patients received treatment, and the treatment was identified in nine cases. In most cases, treatment was short and patients improved almost immediately. Occupational therapy was the most common treatment. Other effective methods were hypnosis (1), relaxation (1), passive movements (2), and probable \"persuasion and re-education\" (6). DISCUSSION The high success rate in treating psychogenic disorders in Hurst's film would be considered impressive by modern standards, and has raised doubt in recent years as to whether parts of the film were staged and/or acted.", "author" : [ { "dropping-particle" : "", "family" : "Moscovich", "given" : "Mariana", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Estupinan", "given" : "Danny", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Qureshi", "given" : "Muhammad", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Okun", "given" : "Michael S", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Tremor and other hyperkinetic movements", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "2013" ] ] }, "page" : "1-7", "title" : "Shell shock: Psychogenic gait and other movement disorders-A film review.", "type" : "article-journal", "volume" : "3" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[26]", "plainTextFormattedCitation" : "[26]", "previouslyFormattedCitation" : "[26]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[26], described facial weakness and ptosis which resolved rapidly with suggestion and persuasionADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Hurst", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1920" ] ] }, "publisher" : "Henry Frowde, Hodder & Stoughton, Oxford University Press", "publisher-place" : "London", "title" : "The psychology of the special senses and their functional disorders", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[23]", "plainTextFormattedCitation" : "[23]", "previouslyFormattedCitation" : "[23]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[23]. Hurst also provides some of the best images of patients with ‘hysterical’ facial spasm (Fig. 2). There were sporadic mentions of 'hysterical' facial spasm in the 1950s,ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Brain", "given" : "R", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1955" ] ] }, "publisher" : "Oxford University Press", "publisher-place" : "London ", "title" : "Diseases of the Nervous System", "type" : "book", "volume" : "5th" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[27]", "plainTextFormattedCitation" : "[27]", "previouslyFormattedCitation" : "[27]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[27] and 1960sADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Abse", "given" : "Wilfred D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "1966" ] ] }, "publisher" : "John Wright & Sons", "publisher-place" : "Bristol", "title" : "Hysteria and Related Mental Disorders", "type" : "book" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[28]", "plainTextFormattedCitation" : "[28]", "previouslyFormattedCitation" : "[28]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[28] but then the problem largely disappeared from view.From the historical review, functional facial movement disorders were positively described as unilateral facial spasm, most commonly presenting with unilateral orbicularis, lower face or platysma contraction. They differed from other types of dystonia by the sustained nature of their contraction. There was general agreement that functional facial weakness, as opposed to muscle overactivity giving the appearance of overactivity, was very rare.Previous published reports of atypical facial movement likely to represent functional facial movement disordersWith hindsight, and as previously described by Fasano et al.ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.25190", "ISSN" : "1531-8257", "PMID" : "23033125", "abstract" : "The facial phenotype of psychogenic movement disorders has not been fully characterized. Seven tertiary-referral movement disorders centers using a standardized data collection on a computerized database performed a retrospective chart review of psychogenic movement disorders involving the face. Patients with organic forms of facial dystonia or any medical or neurological disorder known to affect facial muscles were excluded. Sixty-one patients fulfilled the inclusion criteria for psychogenic facial movement disorders (91.8% females; age: 37.0 \u00b1 11.3 years). Phasic or tonic muscular spasms resembling dystonia were documented in all patients most commonly involving the lips (60.7%), followed by eyelids (50.8%), perinasal region (16.4%), and forehead (9.8%). The most common pattern consisted of tonic, sustained, lateral, and/or downward protrusion of one side of the lower lip with ipsilateral jaw deviation (84.3%). Ipsi- or contralateral blepharospasm and excessive platysma contraction occurred in isolation or combined with fixed lip dystonia (60.7%). Spasms were reported as painful in 24.6% of cases. Symptom onset was abrupt in most cases (80.3%), with at least 1 precipitating psychological stress or trauma identified in 57.4%. Associated body regions involved included upper limbs (29.5%), neck (16.4%), lower limbs (16.4%), and trunk (4.9%). There were fluctuations in severity and spontaneous exacerbations and remissions (60%). Prevalent comorbidities included depression (38.0%) and tension headache (26.4%). Fixed jaw and/or lip deviation is a characteristic pattern of psychogenic facial movement disorders, occurring in isolation or in combination with other psychogenic movement disorders or other psychogenic features.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "Alfonso", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Valadas", "given" : "Anabela", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Prashanth", "given" : "L K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "Anthony E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Munhoz", "given" : "Renato P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Morgante", "given" : "Francesca", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Duker", "given" : "Andrew P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Girlanda", "given" : "Paolo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bentivoglio", "given" : "Anna Rita", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Espay", "given" : "Alberto J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2012", "10" ] ] }, "page" : "1544-51", "title" : "Psychogenic facial movement disorders: clinical features and associated conditions.", "type" : "article-journal", "volume" : "27" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[4]", "plainTextFormattedCitation" : "[4]", "previouslyFormattedCitation" : "[4]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[4], some case reports and series of patients with facial dystonia from the 1980s to the early 2000s may be better classified as a functional movement disorder in terms of variability, associated features and response to treatmentADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "ISSN" : "0022-3050", "PMID" : "3734821", "abstract" : "The clinical features, differential diagnosis and treatment of unilateral spasms of the jaw and masticatory muscles are discussed and illustrated by eight cases of unilateral jaw spasms of various aetiologies. These include focal dystonia of the jaw, hemimasticatory spasm with and without facial hemiatrophy, paroxysmal events in multiple sclerosis and tetany. Attention is particularly drawn to four cases of unilateral dystonia of the jaw which has not been described before.", "author" : [ { "dropping-particle" : "", "family" : "Thompson", "given" : "P D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Obeso", "given" : "J a", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Delgado", "given" : "G", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Gallego", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Marsden", "given" : "C D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of neurology, neurosurgery, and psychiatry", "id" : "ITEM-1", "issue" : "6", "issued" : { "date-parts" : [ [ "1986", "6" ] ] }, "page" : "651-6", "title" : "Focal dystonia of the jaw and the differential diagnosis of unilateral jaw and masticatory spasm.", "type" : "article-journal", "volume" : "49" }, "uris" : [ "" ] }, { "id" : "ITEM-2", "itemData" : { "DOI" : "10.1002/mds.10697", "ISSN" : "0885-3185", "PMID" : "15197704", "abstract" : "Four patients, aged 25 to 42 years presented with acute onset of a movement disorder characterized by a tonic, sustained, lateral and outward protrusion of one half of the lower lip. The movement disorder was present at rest, while in some patients, it was also present during speech. In all cases, the abnormal lip posture could be suppressed voluntarily. Neurological examination was otherwise normal. Extensive laboratory investigation failed to reveal any causative factors for secondary focal dystonia. Treatment with oral medications and botulinum toxin was mostly ineffective. Spontaneous remissions were frequent.", "author" : [ { "dropping-particle" : "", "family" : "Kleopa", "given" : "Kleopas a", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Kyriakides", "given" : "Theodoros", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-2", "issue" : "6", "issued" : { "date-parts" : [ [ "2004", "6" ] ] }, "page" : "663-6", "title" : "A novel movement disorder of the lower lip.", "type" : "article-journal", "volume" : "19" }, "uris" : [ "" ] }, { "id" : "ITEM-3", "itemData" : { "DOI" : "10.1002/mds.20578", "ISSN" : "0885-3185", "PMID" : "16001429", "author" : [ { "dropping-particle" : "", "family" : "Wohlgemuth", "given" : "Marielle", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Pasman", "given" : "Jaco W", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Swart", "given" : "Bert J M", "non-dropping-particle" : "de", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Horstink", "given" : "Martin W I M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-3", "issue" : "8", "issued" : { "date-parts" : [ [ "2005", "8" ] ] }, "page" : "1085-6", "title" : "Movement disorder of the lower lip.", "type" : "article-journal", "volume" : "20" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[29\u201331]", "plainTextFormattedCitation" : "[29\u201331]", "previouslyFormattedCitation" : "[29\u201331]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[29–31]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "PMID" : "11703477", "abstract" : "OBJECTIVE: To describe a patient with facial pain, ipsilateral facial dystonia, and phantom supernumerary teeth, beginning after resection of hypertrophic gums. She had familial sensorineural deafness. BACKGROUND: Atypical cranial dystonia subsequent to dental procedures is associated with facial pain, dysesthesias, or phantom phenomena, but not with phantom supernumerary teeth. Patients with migraine are susceptible to experience cephalic phantom phenomena after dental procedures. Wynne syndrome is a hereditary autosomal dominant disorder characterized by congenital sensorineural deafness and supernumerary teeth. METHODS: Clinical examination, computerized tomography of paranasal sinuses and facial bones, magnetic resonance imaging of the brain, cranial electrophysiological testing, and electroencephalogram. RESULTS: A 52-year-old woman with history of migraine without aura for 40 years exhibited focal right facial involuntary tonic contracture accompanying chronic severe pain over the same area after gum resection. She reported a daily sensation of having two extra upper canine teeth pressing on her tongue, simulating vampire's (\"Dracula's\") teeth. She had high-frequency bilateral sensorineural deafness. Her computerized tomography studies, brain magnetic resonance imaging, and cranial electrophysiological testing were normal. CONCLUSION: This patient with chronic migraine had atypical cranial dystonia beginning after a dental procedure. Her dystonia was complicated by the unusual phenomenon of phantom supernumerary teeth. This condition may be misdiagnosed as atypical or psychogenic facial pain when facial dystonia is localized or subtle and is, therefore, confused with an idiosyncratic gesture or habitual spasm. Her signs and symptoms are reminiscent of Wynne syndrome", "author" : [ { "dropping-particle" : "", "family" : "Jacome", "given" : "D E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Headache", "id" : "ITEM-1", "issue" : "0017-8748 (Print)", "issued" : { "date-parts" : [ [ "2001", "10" ] ] }, "language" : "eng PT - Case Reports PT - Journal Article SB - IM", "note" : "DA - 20011112", "page" : "892-894", "publisher-place" : "Department of Medicine, Franklin Medical Center, Greenfield, Massachusetts, USA", "title" : "Dracula's teeth syndrome", "type" : "article-journal", "volume" : "41" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[32]", "plainTextFormattedCitation" : "[32]", "previouslyFormattedCitation" : "[32]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[32]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.20732", "ISBN" : "0885-3185 (Print)", "ISSN" : "08853185", "PMID" : "16228999", "author" : [ { "dropping-particle" : "", "family" : "Papapetropoulos", "given" : "Spiridon", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Singer", "given" : "Carlos", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement Disorders", "id" : "ITEM-1", "issue" : "1", "issued" : { "date-parts" : [ [ "2006" ] ] }, "page" : "125-126", "title" : "Lower facial and neck dystonia after a cosmetic surgical procedure: Report of a case [3]", "type" : "article-journal", "volume" : "21" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[33]", "plainTextFormattedCitation" : "[33]", "previouslyFormattedCitation" : "[33]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[33]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.21488", "ISBN" : "1531-8257 (Electronic)\\r0885-3185 (Linking)", "ISSN" : "08853185", "PMID" : "17486646", "abstract" : "A 27-year-old woman with bruxism suffered a spider bite (Loxosceles rufescens) on the left cheek that caused severe local cellulitis, facial palsy, and painful hyperesthesia over the two lower trigeminal nerve divisions. Facial but not trigeminal neuropathy improved, and she developed a labial dystonia that only corrected while pressing the right medial incisor. A specially designed maxillary splint that continuously pressed it suppressed both dystonia and related spontaneous firing of motor unit potentials in electromyography. Overstimulation of the contralateral trigeminal territory possibly compensated for the altered left trigeminal nerve input, balanced proprioceptive influences at the central inhibitory-excitatory circuitry, and controlled dystonia.", "author" : [ { "dropping-particle" : "", "family" : "Entrambasaguas", "given" : "Manuel", "non-dropping-particle" : "De", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Plaza-Costa", "given" : "Andr\u00e9s", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Casal", "given" : "Joaqu\u00edn", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Parra", "given" : "Silvia", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement Disorders", "id" : "ITEM-1", "issue" : "9", "issued" : { "date-parts" : [ [ "2007" ] ] }, "page" : "1355-1358", "title" : "Labial dystonia after facial and trigeminal neuropathy controlled with a maxillary splint", "type" : "article-journal", "volume" : "22" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[34]", "plainTextFormattedCitation" : "[34]", "previouslyFormattedCitation" : "[34]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[34]. Four such cases from the paper by Thompson et al. ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "ISSN" : "0022-3050", "PMID" : "3734821", "abstract" : "The clinical features, differential diagnosis and treatment of unilateral spasms of the jaw and masticatory muscles are discussed and illustrated by eight cases of unilateral jaw spasms of various aetiologies. These include focal dystonia of the jaw, hemimasticatory spasm with and without facial hemiatrophy, paroxysmal events in multiple sclerosis and tetany. Attention is particularly drawn to four cases of unilateral dystonia of the jaw which has not been described before.", "author" : [ { "dropping-particle" : "", "family" : "Thompson", "given" : "P D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Obeso", "given" : "J a", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Delgado", "given" : "G", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Gallego", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Marsden", "given" : "C D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of neurology, neurosurgery, and psychiatry", "id" : "ITEM-1", "issue" : "6", "issued" : { "date-parts" : [ [ "1986", "6" ] ] }, "page" : "651-6", "title" : "Focal dystonia of the jaw and the differential diagnosis of unilateral jaw and masticatory spasm.", "type" : "article-journal", "volume" : "49" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[29]", "plainTextFormattedCitation" : "[29]", "previouslyFormattedCitation" : "[29]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[29] are shown in Fig.3 along with two other subsequent reported cases mentioned by Fasano et al. SchragADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/1531-8257(199905)14:3<492::AID-MDS1018>3.0.CO;2-4", "ISSN" : "08853185", "PMID" : "10348476", "abstract" : "It is generally recognized that focal dystonia of the limbs or cervical region and blepharospasm sometimes follow, and in these cases may be caused or triggered by, peripheral injury. However, the association between peripheral injury and lower cranial dystonia is rare. We report eight cases who developed cranial dystonia within hours to months following a dental procedure. One group of five cases, all women, developed atypical dystonia associated with painful paresthesias at the site of dystonia. Two of these five cases had fixed jaw-deviating dystonia, whereas the remaining three had additional tremor and spread of their dystonia to involve the tongue in all three, and the lips and neck in two cases. These five patients are reminiscent of cases of limb causalgia\u2013dystonia syndrome, which occurs after minor peripheral trauma and can spread. The remaining three cases developed more typical cranial dystonia following the dental procedure. There was no family history of dystonia or prior use of neuroleptics in any of the patients. The close association in time and location of the procedure and onset of symptoms suggests that the onset of the dystonia may have been caused by the dental intervention, but whether there is a causal relationship between the dental intervention and the development of the dyskinesias requires further epidemiologic studies.", "author" : [ { "dropping-particle" : "", "family" : "Schrag", "given" : "Anette", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Quinn", "given" : "Niall P.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Marsden", "given" : "C. David", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement Disorders", "id" : "ITEM-1", "issue" : "3", "issued" : { "date-parts" : [ [ "1999" ] ] }, "page" : "492-496", "title" : "Atypical and typical cranial dystonia following dental procedures", "type" : "article-journal", "volume" : "14" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[35]", "plainTextFormattedCitation" : "[35]", "previouslyFormattedCitation" : "[35]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[35] et al reported eight cases who developed cranial dystonia within hours to months following a dental procedure. Two of these eight cases had fixed jaw deviating dystonia, and four had painful dysaesthesia which the authors suggested was similar to the limb causalgia–dystonia syndrome, now described as complex regional pain syndrome. The authors discussed how the fixed nature of the jaw deviation, lack of sensory geste antagoniste, presence of pain and long duration of symptoms without progression to a segmental or Meige syndrome supported the unusual nature of the movement disorder and left open the possibility of a functional/psychogenic movement disorder in these four cases.Figure 2: Published cases of facial dystonia that in hindsight fit better with a diagnosis of functional movement disorder. Cases A-DADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "ISSN" : "0022-3050", "PMID" : "3734821", "abstract" : "The clinical features, differential diagnosis and treatment of unilateral spasms of the jaw and masticatory muscles are discussed and illustrated by eight cases of unilateral jaw spasms of various aetiologies. These include focal dystonia of the jaw, hemimasticatory spasm with and without facial hemiatrophy, paroxysmal events in multiple sclerosis and tetany. Attention is particularly drawn to four cases of unilateral dystonia of the jaw which has not been described before.", "author" : [ { "dropping-particle" : "", "family" : "Thompson", "given" : "P D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Obeso", "given" : "J a", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Delgado", "given" : "G", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Gallego", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Marsden", "given" : "C D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of neurology, neurosurgery, and psychiatry", "id" : "ITEM-1", "issue" : "6", "issued" : { "date-parts" : [ [ "1986", "6" ] ] }, "page" : "651-6", "title" : "Focal dystonia of the jaw and the differential diagnosis of unilateral jaw and masticatory spasm.", "type" : "article-journal", "volume" : "49" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[29]", "plainTextFormattedCitation" : "[29]", "previouslyFormattedCitation" : "[29]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[29] reproduced by permission of BMJ publishing group Ltd. (A) Painful jaw deviation following dental extraction with normal R2 blink response latency. (B) Intermittent left face/tongue/jaw spasm. (C) Episodes of complex face and eye spasm with convergence spams of the left eye. An original diagnosis of multiple sclerosis was made, but MRI brain scan and CSF parameters were normal. (D) Episodes lasting 2-5 minutes as shown associated with hyperventilation and relieved by intravenous calcium gluconate despite normal calcium levels during the attacks. (E) Two of the four cases of acute lip deviation reported by Kleopa et al.ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.10697", "ISSN" : "0885-3185", "PMID" : "15197704", "abstract" : "Four patients, aged 25 to 42 years presented with acute onset of a movement disorder characterized by a tonic, sustained, lateral and outward protrusion of one half of the lower lip. The movement disorder was present at rest, while in some patients, it was also present during speech. In all cases, the abnormal lip posture could be suppressed voluntarily. Neurological examination was otherwise normal. Extensive laboratory investigation failed to reveal any causative factors for secondary focal dystonia. Treatment with oral medications and botulinum toxin was mostly ineffective. Spontaneous remissions were frequent.", "author" : [ { "dropping-particle" : "", "family" : "Kleopa", "given" : "Kleopas a", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Kyriakides", "given" : "Theodoros", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "6", "issued" : { "date-parts" : [ [ "2004", "6" ] ] }, "page" : "663-6", "title" : "A novel movement disorder of the lower lip.", "type" : "article-journal", "volume" : "19" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[30]", "plainTextFormattedCitation" : "[30]", "previouslyFormattedCitation" : "[30]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[30] associated with ipsilateral limb weakness. Reproduced by permission of John Wiley and Sons. (F): One of two similar cases to Kleopa reported by Wohlgemuth et al.ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.20578", "ISSN" : "0885-3185", "PMID" : "16001429", "author" : [ { "dropping-particle" : "", "family" : "Wohlgemuth", "given" : "Marielle", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Pasman", "given" : "Jaco W", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Swart", "given" : "Bert J M", "non-dropping-particle" : "de", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Horstink", "given" : "Martin W I M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "8", "issued" : { "date-parts" : [ [ "2005", "8" ] ] }, "page" : "1085-6", "title" : "Movement disorder of the lower lip.", "type" : "article-journal", "volume" : "20" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[31]", "plainTextFormattedCitation" : "[31]", "previouslyFormattedCitation" : "[31]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[31]. Reproduced by permission of John Wiley and Sons. At the time of Thompson et al.'s paper there remained a common view that 'psychogenic' movement disorder could only be diagnosed in patients with recent life events or with concurrent psychiatric abnormalities. In addition, a diagnosis of hysteria was still seen as pejorative and therefore often avoided in cases where the doctor believed the patient had a genuine problem. In a climate in which some focal dystonias had only recently been 'rescued' from psychodynamic interpretations such as torticollis being a 'turning away from responsibility' it is understandable that there was a desire to avoid the diagnosis of “hysteria”, although David Marsden, who led the work on focal dystonia was perspicacious on hysteria and made the diagnosis oftenADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/B978-0-12-801772-2.00003-5", "ISBN" : "9780128017722", "ISSN" : "00729752", "PMID" : "27719844", "abstract" : "The history of functional neurologic disorders in the 20th century from the point of view of the neurologist is U-shaped. A flurry of interest between the 1880s and early 1920s gave way to lack of interest, skepticism, and concern about misdiagnosis. This was mirrored by increasing professional and geographic divisions between neurology and psychiatry after the First World War. In the 1990s the advent of imaging and other technology highlighted the positive nature of a functional diagnosis. Having been closer in the early 20th century but later more separate, these disorders are now once again the subject of academic and clinical interest, although arguably still very much on the fringes of neurology and neuropsychiatry. Revisiting older material provides a rich source of ideas and data for today's clinical researcher, but also offers cautionary tales of theories and treatments that led to stagnation rather than advancement of the field. Patterns of treatment do have a habit of repeating themselves, for example, the current enthusiasm for transcranial magnetic stimulation compared to the excitement about electrotherapy in the 19th century. For these reasons, an understanding of the history of functional disorders in neurology is arguably more important than it is for other areas of neurologic practice.", "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "J.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Handbook of Clinical Neurology", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "2017" ] ] }, "page" : "25-36", "title" : "Chapter 3 \u2013 Neurologic approaches to hysteria, psychogenic and functional disorders from the late 19th century onwards", "type" : "chapter", "volume" : "139" }, "uris" : [ "" ] }, { "id" : "ITEM-2", "itemData" : { "PMID" : "3726004", "author" : [ { "dropping-particle" : "", "family" : "Marsden", "given" : "C D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Psychol.Med.", "id" : "ITEM-2", "issue" : "2", "issued" : { "date-parts" : [ [ "1986", "5" ] ] }, "page" : "277-288", "title" : "Hysteria - a neurologist's view", "type" : "article-journal", "volume" : "16" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[8,36]", "plainTextFormattedCitation" : "[8,36]", "previouslyFormattedCitation" : "[8,36]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[8,36]. The problems with diagnosis at that time can be seen, for example, in the four patients said to have psychogenic blepharospasm in the paper by Cavenar et al in 1978ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Cavenar", "given" : "Jesse O", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issue" : "18", "issued" : { "date-parts" : [ [ "2017" ] ] }, "title" : "Blepharospasm : Organic or functional ? Recommended articles Blinking and essential blepharospasm Meige ' s syndrome : A neuropsychiatric disord \u2026 Activation of GPR30 attenuates chronic pain \u2026 Citing articles ( 18 ) Related book content", "type" : "article-journal" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[37]", "plainTextFormattedCitation" : "[37]", "previouslyFormattedCitation" : "[37]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[37]. based on profound psychopathology. All of these patients had features in keeping with organic blepharospasm with bilateral involvement and psychiatric comorbidity. The changing view of functional movement disorders now encompasses movement disorders that are genuine, variable, utilise voluntary muscle pathways and have diagnostic features that indicate the role that attentional focus plays in the movements. A diagnosis of functional movement disorder no longer requires psychological causation in the latest revision of DSM-5, the American Psychiatric Association’s psychiatric classificationADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/j.jpsychores.2011.07.005", "ISSN" : "1879-1360", "PMID" : "22118377", "abstract" : "Conversion disorder in the Diagnostic and Statistical Manual of Mental Disorders (DSM-IV-TR) describes neurological symptoms, including weakness, numbness and events resembling epilepsy or syncope, which can be positively identified as not being due to recognised neurological disease. This review combines perspectives from psychiatry, psychology and neurology to identify and discuss key problems with the current diagnostic DSM-IV criteria for conversion disorder and to make the following proposals for DSM-5: (a) abandoning the label \"conversion disorder\" and replacing it with an alternative term that is both theoretically neutral and potentially more acceptable to patients and practitioners; (b) relegating the requirements for \"association of psychological factors\" and the \"exclusion of feigning\" to the accompanying text; (c) adding a criterion requiring clinical findings of internal inconsistency or incongruity with recognised neurological or medical disease and altering the current 'disease exclusion' criteria to one in which the symptom must not be 'better explained' by a disease if present, (d) adding a 'cognitive symptoms' subtype. We also discuss whether conversion symptoms are better classified with other somatic symptom disorders or with dissociative disorders and how we might address the potential heterogeneity of conversion symptoms in classification.", "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "Jon", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "LaFrance", "given" : "W. Curt", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Brown", "given" : "Richard", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Spiegel", "given" : "David", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Levenson", "given" : "James L.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Sharpe", "given" : "Michael", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "LaFrance Jr.", "given" : "W C", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Brown", "given" : "Richard", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Spiegel", "given" : "David", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Levenson", "given" : "James L.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Sharpe", "given" : "Michael", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "LaFrance", "given" : "W. Curt", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Brown", "given" : "Richard", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Spiegel", "given" : "David", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Levenson", "given" : "James L.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Sharpe", "given" : "Michael", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of psychosomatic research", "id" : "ITEM-1", "issue" : "6", "issued" : { "date-parts" : [ [ "2011", "12" ] ] }, "language" : "eng PT - Journal Article SB - IM", "page" : "369-376", "publisher" : "Elsevier Inc.", "publisher-place" : "Dept Clinical Neurosciences, University of Edinburgh, Western General Hospital, Crewe Rd, Edinburgh EH4 2XU, UK. Jon.Stone@ed.ac.uk", "title" : "Conversion disorder: current problems and potential solutions for DSM-5.", "type" : "article-journal", "volume" : "71" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[38]", "plainTextFormattedCitation" : "[38]", "previouslyFormattedCitation" : "[38]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[38].Emergence of reports of functional (psychogenic) facial movement disordersKeane, in 1986, noted the presence of 'wrong way tongue deviation' as a sign of functional disorder and commented on the presence of ptosis in some patientsADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "PMID" : "3762952", "abstract" : "Strong deviation of the protruded tongue towards the \"normal\" side is an old sign of hysterical hemiparesis. The following four cases illustrate that hysterical tongue deviation continues to offer a valuable diagnostic clue", "author" : [ { "dropping-particle" : "", "family" : "Keane", "given" : "J R", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Neurology", "id" : "ITEM-1", "issue" : "0028-3878 (Print)", "issued" : { "date-parts" : [ [ "1986", "10" ] ] }, "language" : "eng PT - Case Reports PT - Journal Article SB - AIM SB - IM", "note" : "DA - 19861103", "page" : "1406-1407", "title" : "Wrong-way deviation of the tongue with hysterical hemiparesis", "type" : "article-journal", "volume" : "36" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[39]", "plainTextFormattedCitation" : "[39]", "previouslyFormattedCitation" : "[39]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[39]. Case reports of functional or psychogenic pseudoptosis (in fact related to orbicularis oculi contraction rather than eyelid drooping) appeared from 1997 onwards ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "abstract" : "Three patients with psychogenic pseudoptosis of one eyelid are reported. All showed depression of the eyebrow on the affected side. The clinical course varied: in two patients the symptom resolved spontaneously after positive reassurance; in the third patient it remained unchanged for 2 years", "author" : [ { "dropping-particle" : "", "family" : "Hop", "given" : "J W", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Frijns", "given" : "C J M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "Van", "family" : "Gijn", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Gijn", "given" : "J", "non-dropping-particle" : "van", "parse-names" : false, "suffix" : "" } ], "container-title" : "J.Neurol.", "id" : "ITEM-1", "issue" : "10", "issued" : { "date-parts" : [ [ "1997", "10" ] ] }, "note" : " From Duplicate 2 ( Psychogenic pseudoptosis - Hop, J W; Frijns, C J; van Gijn, J )\n \n98066157\n \n ", "page" : "623-624", "publisher-place" : "University Department of Neurology, Utrecht, The Netherlands. j.w.hop@neuro.azu.nl", "title" : "Psychogenic pseudoptosis", "type" : "article-journal", "volume" : "244" }, "uris" : [ "" ] }, { "id" : "ITEM-2", "itemData" : { "DOI" : "10.1046/j.1474-7766.2002.00103.x", "ISSN" : "14747758", "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "J.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Practical Neurology", "id" : "ITEM-2", "issue" : "6", "issued" : { "date-parts" : [ [ "2002" ] ] }, "title" : "Pseudo-ptosis", "type" : "article-journal", "volume" : "2" }, "uris" : [ "" ] }, { "id" : "ITEM-3", "itemData" : { "DOI" : "10.1097/IOP.0000000000000069", "ISBN" : "0000000000000", "ISSN" : "0740-9303", "PMID" : "24608330", "abstract" : "Conversion disorders are among common psychological problems. Pseudoptosis is an uncommon presentation of this category of disorders. Herein, the authors report 2 cases of psychogenic pseudoptosis. The first case is a 21-year-old man who presented with sudden onset of ptosis in his OS 2 weeks previous. The patient reported episodes of left upper eyelid drooping during the last 7 months. The second case is a 10-year-old girl with ptosis onset 6 months previously, after a viral conjunctivitis. In both patients, specific presentations in favor of nonorganic ptosis were observed; all neurological and paraclinical evaluations were normal, and considerable familial or social stressors were found. Both patients received psychological treatment, and no recurrence was observed afterward.", "author" : [ { "dropping-particle" : "", "family" : "Bagheri", "given" : "Abbas", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Abbasnia", "given" : "Ehsan", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Pakravan", "given" : "Mohammad", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Roshani", "given" : "Maryam", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tavakoli", "given" : "Mehdi", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Ophthalmic Plastic and Reconstructive Surgery", "id" : "ITEM-3", "issue" : "3", "issued" : { "date-parts" : [ [ "2015", "3" ] ] }, "page" : "e55-e57", "title" : "Psychogenic Unilateral Pseudoptosis", "type" : "article-journal", "volume" : "31" }, "uris" : [ "", "" ] }, { "id" : "ITEM-4", "itemData" : { "DOI" : "10.1016/j.pediatrneurol.2009.06.006", "ISSN" : "1873-5150", "PMID" : "19818939", "abstract" : "A 13- year-old girl presented with unilateral psychogenic pseudoptosis. She had sudden-onset ptosis on the left side 3 weeks before presentation. Careful review of her medical history revealed multiple assessments for chronic pain symptoms over the previous 2 years. Physical examination revealed isolated ptosis on the left side, with no other abnormal neurologic findings. On sustained upward gaze, mild elevation of the upper eyelid accompanied by slight elevation of the lower eye lid was seen. Imaging of the brain, laboratory investigations and Electrophysiologic studies for neurogenic and myogenic pathologies were normal. Dramatic and sustained response with complete resolution of ptosis was observed after administration of placebo during the edrophonium test, thus confirming the diagnosis of psychogenic pseudoptosis. The patient was offered psychologic assessment and support. There was no recurrence of symptoms at follow-up after 2 months. Psychogenic pseudoptosis is an uncommon manifestation of conversion disorder in children. Early recognition of the condition and institution of appropriate psychologic therapeutic interventions are essential for good recovery.", "author" : [ { "dropping-particle" : "", "family" : "Peer Mohamed", "given" : "Basheer A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Patil", "given" : "Shekhar G", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Pediatric neurology", "id" : "ITEM-4", "issue" : "5", "issued" : { "date-parts" : [ [ "2009", "11" ] ] }, "page" : "364-6", "publisher" : "Elsevier Inc.", "title" : "Psychogenic unilateral pseudoptosis.", "type" : "article-journal", "volume" : "41" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[40\u201343]", "plainTextFormattedCitation" : "[40\u201343]", "previouslyFormattedCitation" : "[40\u201343]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[40–43]. Psychogenic hemifacial spasm, involving the lower face, was reported by Tan & Jankovic in 2001 [n=4]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "abstract" : "Facial spasms that distort facial expression are typically due to facial dystonia, tics, and hemifacial spasm (HFS). Psychogenic facial spasms, however, have not been well characterized. The authors sought to 1) determine prevalence of psychogenic facial spasm in patients referred for evaluation of HFS and 2) draw attention to clinical characteristics and potential diagnostic pitfalls. Among 210 consecutive patients referred for evaluation of HFS, 5 (2.4%) received diagnoses of psychogenic facial spasm. All patients were female; mean age was 34.6 years (range 26-45) and mean symptom duration 1.1 years (range 2 wk-2 yr). Onset was left-sided in 3 patients, and the lid was the initial site affected in 2 patients. This series of patients shows that facial spasms, although usually of neurovascular etiology, may be the initial or only manifestation of a psychogenic movement disorder, often associated with an underlying depression.", "author" : [ { "dropping-particle" : "", "family" : "Tan", "given" : "E K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Jankovic", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "The Journal of neuropsychiatry and clinical neurosciences", "id" : "ITEM-1", "issue" : "3", "issued" : { "date-parts" : [ [ "2001" ] ] }, "page" : "380-384", "title" : "Psychogenic hemifacial spasm.", "type" : "article-journal", "volume" : "13" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[1]", "plainTextFormattedCitation" : "[1]", "previouslyFormattedCitation" : "[1]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[1], with subsequent case series by Tarsy et al. [n=5]ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Dengenhardt", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Zadikoff", "given" : "C", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Psychogenic Movement Disorders", "editor" : [ { "dropping-particle" : "", "family" : "Hallett", "given" : "M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Jankovic", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "AE", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Cloninger", "given" : "CR", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Yudofksy", "given" : "SC", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "2006" ] ] }, "page" : "341-3", "publisher" : "Lippincott Williams & Wilkins", "publisher-place" : "Philadelphia", "title" : "Psychogenic facial spasm (the smirk) presenting as hemifacial spasm", "type" : "chapter" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[2]", "plainTextFormattedCitation" : "[2]", "previouslyFormattedCitation" : "[2]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[2], and a report by Stone [n=1ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Carson", "given" : "A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of Neurology, Neurosurgery & Psychiatry", "id" : "ITEM-1", "issue" : "10", "issued" : { "date-parts" : [ [ "2010" ] ] }, "page" : "e8--e9", "publisher" : "BMJ Publishing Group Ltd", "title" : "020 Psychogenic/dissociative/functional facial symptoms\u2014a case report", "type" : "article-journal", "volume" : "81" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[3]", "plainTextFormattedCitation" : "[3]", "previouslyFormattedCitation" : "[3]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[3]] between 2001-2010. Patients with psychogenic blepharospasm were also reported in case series of other psychogenic movement disorders without detailed description of how the positive diagnosis had been madeADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "ISSN" : "0022-3050", "PMID" : "7561921", "abstract" : "Of 842 consecutive patients with movement disorders seen over a 71 month period, 28 (3.3%) were diagnosed as having a documented or clinically established psychogenic movement disorder. Tremor was most common (50%) followed by dystonia, myoclonus, and parkinsonism. Clinical descriptions of various types are reviewed. Clinical characteristics common in these patients included distractability (86%), abrupt onset (54%), and selective disabilities (39%). Distractability seems to be most important in tremor and least important in dystonia. Other diagnostic clues included entrainment of tremor to the frequency of repetitive movements of another limb, fatigue of tremor, stimulus sensitivity, and previous history of psychogenic illness. On examination, 71% had other psychogenic features. Over 60% had a clear history of a precipitating event and secondary gain and 50% had a psychiatric diagnosis (usually depression). Twenty five per cent of patients presented with combined psychogenic movement disorder and organic movement disorder; 35% resolved and this subgroup had a shorter duration of disease than those who are unresolved. Psychogenic movement disorder represents an uncommon diagnosis among patients with movement disorders. The ability to make a diagnosis rests on the presence of a multitude of clinical clues and therapeutic action should be taken as early as possible.", "author" : [ { "dropping-particle" : "", "family" : "Factor", "given" : "S A", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Podskalny", "given" : "G D", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Molho", "given" : "E S", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of neurology, neurosurgery, and psychiatry", "id" : "ITEM-1", "issue" : "4", "issued" : { "date-parts" : [ [ "1995", "10" ] ] }, "language" : "eng PT - Journal Article PT - Research Support, Non-U.S. Gov't SB - IM", "note" : "DA - 19951103", "page" : "406-12", "publisher-place" : "Albany Medical College, Department of Neurology, New York 12208, USA", "title" : "Psychogenic movement disorders: frequency, clinical profile, and characteristics.", "type" : "article-journal", "volume" : "59" }, "uris" : [ "" ] }, { "id" : "ITEM-2", "itemData" : { "ISSN" : "0091-3952", "PMID" : "7872143", "author" : [ { "dropping-particle" : "", "family" : "Williams", "given" : "D T", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Ford", "given" : "B", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Fahn", "given" : "S", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Advances in neurology", "id" : "ITEM-2", "issued" : { "date-parts" : [ [ "1995" ] ] }, "language" : "eng PT - Case Reports PT - Journal Article PT - Research Support, Non-U.S. Gov't PT - Review SB - IM", "note" : "DA - 19950327", "page" : "231-57", "publisher-place" : "Department of Psychiatry, Columbia-Presbyterian Medical Center, New York, New York 10032", "title" : "Phenomenology and psychopathology related to psychogenic movement disorders.", "type" : "article-journal", "volume" : "65" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[44,45]", "plainTextFormattedCitation" : "[44,45]", "previouslyFormattedCitation" : "[44,45]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[44,45]. In 2011 Schwingenschuh and colleagues reported that the blink recovery cycle reflex was normal in patients with "presumed psychogenic" blepharospasm (n=9) but abnormal in essential blepharospasm ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1212/WNL.0b013e31820c3074", "ISBN" : "1526-632X (Electronic)\\n0028-3878 (Linking)", "ISSN" : "00283878", "PMID" : "21321334", "abstract" : "Psychogenic blepharospasm is difficult to distinguish clinically from benign essential blepharospasm (BEB). The blink reflex recovery cycle measures the excitability of human brainstem interneurons and is abnormal in BEB. We wished to study the blink reflex recovery cycle in patients with atypical (presumed psychogenic) blepharospasm (AB).", "author" : [ { "dropping-particle" : "", "family" : "Schwingenschuh", "given" : "P.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Katschnig", "given" : "P.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Edwards", "given" : "M. J.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Teo", "given" : "J. T H", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Korlipara", "given" : "L. V P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Rothwell", "given" : "J. C.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "K. P.", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Neurology", "id" : "ITEM-1", "issue" : "1526-632X (Electronic)", "issued" : { "date-parts" : [ [ "2011", "2" ] ] }, "language" : "eng PT - Journal Article PT - Research Support, Non-U.S. Gov't SB - AIM SB - IM", "page" : "610-614", "publisher-place" : "UCL Institute of Neurology, London WC1N 3BG, UK", "title" : "The blink reflex recovery cycle differs between essential and presumed psychogenic blepharospasm", "type" : "article-journal", "volume" : "76" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[46]", "plainTextFormattedCitation" : "[46]", "previouslyFormattedCitation" : "[46]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[46]. More recently Ganos et al describe psychogenic paroxysmal movement disorders affecting the face and head in six and seven patients respectivelyADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/j.parkreldis.2013.09.012", "ISSN" : "1873-5126", "PMID" : "24090947", "abstract" : "BACKGROUND: The diagnosis of psychogenic paroxysmal movement disorders (PPMD) can be challenging, in particular their distinction from the primary paroxysmal dyskinesias (PxD) remains difficult.\\n\\nMETHODS: Here we present a large series of 26 PPMD cases, describe their characteristics, contrast them with primary PxD and focus on their distinguishing diagnostic features.\\n\\nRESULTS: Mean age at onset was 38.6 years, i.e. much later than primary PxD. Women were predominantly affected (73%). Most subjects (88.4%) had long attacks, and unlike primary PxD there was a very high within-subject variability for attack phenomenology, duration and frequency. Dystonia was the most common single movement disorder presentation, but 69.2% of the patients had mixed or complex PxD. In 50% of PPMD cases attack triggers could be identified but these were unusual for primary PxD. 42.3% of patients employed unusual strategies to alleviate or stop the attacks. Response to typical medication used for primary PxD was poor. Precipitation of the disorder due to physical or emotional life events and stressors were documented in 57.6% and 65.3% of the cases respectively. Additional interictal psychogenic signs were documented in 34.6% and further medically unexplained somatic symptoms were present in 50% of the cases. 19.2% of patients had a comorbid organic movement disorder and 26.9% had pre-existing psychiatric comorbidities.\\n\\nCONCLUSION: Although the phenotypic presentation of PPMD can be highly diverse, certain clinical characteristics help in distinguishing this condition from the primary forms of PxD. Recognition is important as multidisciplinary treatment approaches led to significant improvement in most cases.", "author" : [ { "dropping-particle" : "", "family" : "Ganos", "given" : "Christos", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Aguirregomozcorta", "given" : "Maria", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Batla", "given" : "Amit", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Stamelou", "given" : "Maria", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Schwingenschuh", "given" : "Petra", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "M\u00fcnchau", "given" : "Alexander", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Edwards", "given" : "Mark J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Parkinsonism & related disorders", "id" : "ITEM-1", "issue" : "1", "issued" : { "date-parts" : [ [ "2013" ] ] }, "page" : "41-46", "publisher" : "Elsevier Ltd", "title" : "Psychogenic paroxysmal movement disorders - Clinical features and\u00a0diagnostic clues.", "type" : "article-journal", "volume" : "20" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[47]", "plainTextFormattedCitation" : "[47]", "previouslyFormattedCitation" : "[47]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[47]. Ganos et al also described another case with bilateral tonic closure of both eyes including video in a review article on functional dystoniaADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mdc3.12013", "author" : [ { "dropping-particle" : "", "family" : "Ganos", "given" : "Christos", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Edwards", "given" : "Mark J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "id" : "ITEM-1", "issue" : "February", "issued" : { "date-parts" : [ [ "2014" ] ] }, "page" : "36-44", "title" : "The Phenomenology of Functional ( Psychogenic ) Dystonia Phenotypic Characteristics of FD", "type" : "article-journal" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[48]", "plainTextFormattedCitation" : "[48]", "previouslyFormattedCitation" : "[48]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[48]. The case reports of Gozke et al ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "author" : [ { "dropping-particle" : "", "family" : "G\u00f6zke", "given" : "Eren", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Sara\u00e7o\u011flu", "given" : "Boran Can", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Eser", "given" : "Mustafa", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Reyhani", "given" : "Aylin", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Ak", "given" : "Pelin Do\u011fan", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "International journal of clinical neurosciences and mental health", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "2016" ] ] }, "title" : "Psychogenic facial movement disorder : a case report", "type" : "article-journal" }, "uris" : [ "", "" ] } ], "mendeley" : { "formattedCitation" : "[49]", "plainTextFormattedCitation" : "[49]", "previouslyFormattedCitation" : "[49]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[49] and illustrated case series of four patients with tonic lip deviation by Colosimo et al highlight the growing awareness of functional facial movement disorders ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/j.jns.2015.08.1552", "ISSN" : "0022510X", "author" : [ { "dropping-particle" : "", "family" : "Colosimo", "given" : "Carlo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of the Neurological Sciences", "id" : "ITEM-1", "issued" : { "date-parts" : [ [ "2015" ] ] }, "publisher" : "Elsevier B.V.", "title" : "Tonic lip deviation as a distinctive form of psychogenic facial dystonia", "type" : "article-journal" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[50]", "plainTextFormattedCitation" : "[50]", "previouslyFormattedCitation" : "[50]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[50].New case series in contextData from our new case series, the second largest, are described in table one and compared to the other large series of patients with functional facial movement disorders described by Fasano et alADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.25190", "ISSN" : "1531-8257", "PMID" : "23033125", "abstract" : "The facial phenotype of psychogenic movement disorders has not been fully characterized. Seven tertiary-referral movement disorders centers using a standardized data collection on a computerized database performed a retrospective chart review of psychogenic movement disorders involving the face. Patients with organic forms of facial dystonia or any medical or neurological disorder known to affect facial muscles were excluded. Sixty-one patients fulfilled the inclusion criteria for psychogenic facial movement disorders (91.8% females; age: 37.0 \u00b1 11.3 years). Phasic or tonic muscular spasms resembling dystonia were documented in all patients most commonly involving the lips (60.7%), followed by eyelids (50.8%), perinasal region (16.4%), and forehead (9.8%). The most common pattern consisted of tonic, sustained, lateral, and/or downward protrusion of one side of the lower lip with ipsilateral jaw deviation (84.3%). Ipsi- or contralateral blepharospasm and excessive platysma contraction occurred in isolation or combined with fixed lip dystonia (60.7%). Spasms were reported as painful in 24.6% of cases. Symptom onset was abrupt in most cases (80.3%), with at least 1 precipitating psychological stress or trauma identified in 57.4%. Associated body regions involved included upper limbs (29.5%), neck (16.4%), lower limbs (16.4%), and trunk (4.9%). There were fluctuations in severity and spontaneous exacerbations and remissions (60%). Prevalent comorbidities included depression (38.0%) and tension headache (26.4%). Fixed jaw and/or lip deviation is a characteristic pattern of psychogenic facial movement disorders, occurring in isolation or in combination with other psychogenic movement disorders or other psychogenic features.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "Alfonso", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Valadas", "given" : "Anabela", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Prashanth", "given" : "L K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "Anthony E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Munhoz", "given" : "Renato P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Morgante", "given" : "Francesca", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Duker", "given" : "Andrew P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Girlanda", "given" : "Paolo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bentivoglio", "given" : "Anna Rita", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Espay", "given" : "Alberto J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2012", "10" ] ] }, "page" : "1544-51", "title" : "Psychogenic facial movement disorders: clinical features and associated conditions.", "type" : "article-journal", "volume" : "27" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[4]", "plainTextFormattedCitation" : "[4]", "previouslyFormattedCitation" : "[4]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[4]. The presence of 41 cases collected over a 5-year period indicates a movement disorder that must be relatively common relative to many other disorders. The neurologist seeing these patients (JS) does have an interest in functional disorders but nevertheless many of these patients were seen in a general neurological setting and the population catchment of the centre is only 1 million persons.Patients in our case series were predominantly female (81%) and middle aged (mean age 44yrs). The most common signs were downward lip pulling and orbicularis oculis spasm (both 90%). Platysmal overactivity frequently accompanied this (85%). It was usually unilateral (90%), without right or left preponderance. 91% were paroxysmal by which we mean episodes of spasm that may last seconds to minutes before resolving. The rest had a persistent presentation where some degree of spasm was present most or nearly all the time. This was higher than the frequency in the Fasano et al series (60%). Tongue deviation, exclusively towards the side of facial spasm, was not seen commonly (12%) and jaw deviation was much less frequently seen than was described by Fasano et al (22% vs. 84%). Jaw deviation was assessed by inspection and not measured in a systematic way. This subjective assessment may explain the difference in data.Not recorded by Fasano et alADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.25190", "ISSN" : "1531-8257", "PMID" : "23033125", "abstract" : "The facial phenotype of psychogenic movement disorders has not been fully characterized. Seven tertiary-referral movement disorders centers using a standardized data collection on a computerized database performed a retrospective chart review of psychogenic movement disorders involving the face. Patients with organic forms of facial dystonia or any medical or neurological disorder known to affect facial muscles were excluded. Sixty-one patients fulfilled the inclusion criteria for psychogenic facial movement disorders (91.8% females; age: 37.0 \u00b1 11.3 years). Phasic or tonic muscular spasms resembling dystonia were documented in all patients most commonly involving the lips (60.7%), followed by eyelids (50.8%), perinasal region (16.4%), and forehead (9.8%). The most common pattern consisted of tonic, sustained, lateral, and/or downward protrusion of one side of the lower lip with ipsilateral jaw deviation (84.3%). Ipsi- or contralateral blepharospasm and excessive platysma contraction occurred in isolation or combined with fixed lip dystonia (60.7%). Spasms were reported as painful in 24.6% of cases. Symptom onset was abrupt in most cases (80.3%), with at least 1 precipitating psychological stress or trauma identified in 57.4%. Associated body regions involved included upper limbs (29.5%), neck (16.4%), lower limbs (16.4%), and trunk (4.9%). There were fluctuations in severity and spontaneous exacerbations and remissions (60%). Prevalent comorbidities included depression (38.0%) and tension headache (26.4%). Fixed jaw and/or lip deviation is a characteristic pattern of psychogenic facial movement disorders, occurring in isolation or in combination with other psychogenic movement disorders or other psychogenic features.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "Alfonso", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Valadas", "given" : "Anabela", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Prashanth", "given" : "L K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "Anthony E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Munhoz", "given" : "Renato P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Morgante", "given" : "Francesca", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Duker", "given" : "Andrew P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Girlanda", "given" : "Paolo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bentivoglio", "given" : "Anna Rita", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Espay", "given" : "Alberto J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2012", "10" ] ] }, "page" : "1544-51", "title" : "Psychogenic facial movement disorders: clinical features and associated conditions.", "type" : "article-journal", "volume" : "27" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[4]", "plainTextFormattedCitation" : "[4]", "previouslyFormattedCitation" : "[4]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[4] but seen in our series, were facial spasms triggered by examination of eye movements or by asking the patients to sustain muscular contraction of the face (51%). This is a similar mechanism to the triggering of functional convergence spasm by sustained gaze in Kaski et al’s study of patients with functional eye movement disorders and likely relates to the effect of sustained attention in functional facial movement disordersADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1016/S1474-4422(15)00226-4", "ISSN" : "14744465", "PMID" : "26581970", "abstract" : "Functional (psychogenic) neurological symptoms are frequently encountered in neurological practice. Cranial movement disorders-affecting the eyes, face, jaw, tongue, or palate-are an under-recognised feature of patients with functional symptoms. They can present in isolation or in the context of other functional symptoms; in particular, for functional eye movements, positive clinical signs such as convergence spasms can be triggered by the clinical examination. Although the specialty of functional neurological disorders has expanded, appreciation of cranial functional movement disorders is still insufficient. Identification of the positive features of cranial functional movement disorders such as convergence and unilateral platysmal spasm might lend diagnostic weight to a suspected functional neurological disorder. Understanding of the differential diagnosis, which is broad and includes many organic causes (eg, stroke), is essential to make an early and accurate diagnosis to prevent complications and initiate appropriate management. Increased understanding of these disorders is also crucial to drive clinical trials and studies of individually tailored therapies.", "author" : [ { "dropping-particle" : "", "family" : "Kaski", "given" : "Diego", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bronstein", "given" : "Adolfo M", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Edwards", "given" : "Mark J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Stone", "given" : "Jon", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "The Lancet Neurology", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2015" ] ] }, "page" : "1196-1205", "title" : "Cranial functional (psychogenic) movement disorders", "type" : "article", "volume" : "14" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[6]", "plainTextFormattedCitation" : "[6]", "previouslyFormattedCitation" : "[6]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[6]. From a clinical perspective we found this a good way of inducing the symptom in those patients who attended clinic with a history of episodic spasm but without the movement disorder at the time of assessment. In 78% patients, there was evidence of functional limb weakness. This was mostly in the ipsilateral limb (91% of the time when present). Our series is also notable for a high frequency of other comorbid functional neurological disorders not reported by Fasano et al including convergence spasm of eye abduction (22%), dissociative (non-epileptic seizures) (32%) and functional dysphonia (12%). Other physical comorbidities including persistent fatigue (83%) and migraine (51%) were also common, in keeping with findings of multiple comorbidity in other functional movement disorder.Migraine triggered functional facial movement disorders in 17% and 7% of patients reported premonitory symptoms that were abolished by paroxysmal movements. Such premonitory symptoms also occur in ‘organic’ forms of oromandibular dystonia and are certainly not diagnosticADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.10063", "ISBN" : "0885-3185", "ISSN" : "08853185", "PMID" : "11836760", "author" : [ { "dropping-particle" : "", "family" : "Berardelli", "given" : "Alfredo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Curra", "given" : "Antonio", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement Disorders", "id" : "ITEM-1", "issue" : "SUPPL. 2", "issued" : { "date-parts" : [ [ "2002" ] ] }, "page" : "70-74", "title" : "Pathophysiology and treatment of cranial dystonia", "type" : "article-journal", "volume" : "17" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[51]", "plainTextFormattedCitation" : "[51]", "previouslyFormattedCitation" : "[51]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[51]. The phenomenology here was similar to patients with non-epileptic attacks who commonly experience an unpleasant aura that is relieved by the attack (even though the attack itself is also unwelcome) ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1136/jnnp-2012-304842", "ISBN" : "2012304842", "ISSN" : "1468-330X", "PMID" : "23538071", "author" : [ { "dropping-particle" : "", "family" : "Stone", "given" : "Jon", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Carson", "given" : "Alan J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Journal of neurology, neurosurgery, and psychiatry", "id" : "ITEM-1", "issue" : "7", "issued" : { "date-parts" : [ [ "2013", "7" ] ] }, "page" : "822-4", "title" : "The unbearable lightheadedness of seizing: wilful submission to dissociative (non-epileptic) seizures.", "type" : "article-journal", "volume" : "84" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[52]", "plainTextFormattedCitation" : "[52]", "previouslyFormattedCitation" : "[52]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[52]. In the context of a functional facial movement disorder this finding could be used in treatment strategies during cognitive behavioural therapy.ConclusionFunctional facial dystonia or spasm has recently been recognised as a relatively common form of functional (psychogenic) movement disorder with clearly identifiable clinical features. We have highlighted historical and 'forgotten' neurological literature dating back to 1887 indicating that it was a well described phenomenon at that time, although awareness of it slipped from general neurological awareness in the middle of the 20th century. With hindsight, many published cases of facial dystonia from the 1980s onwards fit best with this entity. The consistency of presentation over time, especially when the symptom had essentially been lost from view, and which has been reported in many different parts of the world in a similar way, argues for this being a genuine expression of human pathophysiology. Our own case series of 41 patients from one regional centre highlights that this must be a relatively common clinical problem and highlights some new data, especially in relation to triggering manoeuvres during examination and comorbidities with other functional disorders. Documentation of author rolesJ Stone conceived of the project, collected the patient data, started and edited the manuscript. I Hoeritzauer and L Tesolin wrote and edited the manuscript. A Carson organised, critiqued and reviewed the manuscript. Table 1 Clinical features in a new series of 41 patients with functional facial movement disorder. Compared to other large series of Fasano et al.ADDIN CSL_CITATION { "citationItems" : [ { "id" : "ITEM-1", "itemData" : { "DOI" : "10.1002/mds.25190", "ISSN" : "1531-8257", "PMID" : "23033125", "abstract" : "The facial phenotype of psychogenic movement disorders has not been fully characterized. Seven tertiary-referral movement disorders centers using a standardized data collection on a computerized database performed a retrospective chart review of psychogenic movement disorders involving the face. Patients with organic forms of facial dystonia or any medical or neurological disorder known to affect facial muscles were excluded. Sixty-one patients fulfilled the inclusion criteria for psychogenic facial movement disorders (91.8% females; age: 37.0 \u00b1 11.3 years). Phasic or tonic muscular spasms resembling dystonia were documented in all patients most commonly involving the lips (60.7%), followed by eyelids (50.8%), perinasal region (16.4%), and forehead (9.8%). The most common pattern consisted of tonic, sustained, lateral, and/or downward protrusion of one side of the lower lip with ipsilateral jaw deviation (84.3%). Ipsi- or contralateral blepharospasm and excessive platysma contraction occurred in isolation or combined with fixed lip dystonia (60.7%). Spasms were reported as painful in 24.6% of cases. Symptom onset was abrupt in most cases (80.3%), with at least 1 precipitating psychological stress or trauma identified in 57.4%. Associated body regions involved included upper limbs (29.5%), neck (16.4%), lower limbs (16.4%), and trunk (4.9%). There were fluctuations in severity and spontaneous exacerbations and remissions (60%). Prevalent comorbidities included depression (38.0%) and tension headache (26.4%). Fixed jaw and/or lip deviation is a characteristic pattern of psychogenic facial movement disorders, occurring in isolation or in combination with other psychogenic movement disorders or other psychogenic features.", "author" : [ { "dropping-particle" : "", "family" : "Fasano", "given" : "Alfonso", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Valadas", "given" : "Anabela", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bhatia", "given" : "Kailash P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Prashanth", "given" : "L K", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Lang", "given" : "Anthony E", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Munhoz", "given" : "Renato P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Morgante", "given" : "Francesca", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Tarsy", "given" : "Daniel", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Duker", "given" : "Andrew P", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Girlanda", "given" : "Paolo", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Bentivoglio", "given" : "Anna Rita", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" }, { "dropping-particle" : "", "family" : "Espay", "given" : "Alberto J", "non-dropping-particle" : "", "parse-names" : false, "suffix" : "" } ], "container-title" : "Movement disorders : official journal of the Movement Disorder Society", "id" : "ITEM-1", "issue" : "12", "issued" : { "date-parts" : [ [ "2012", "10" ] ] }, "page" : "1544-51", "title" : "Psychogenic facial movement disorders: clinical features and associated conditions.", "type" : "article-journal", "volume" : "27" }, "uris" : [ "" ] } ], "mendeley" : { "formattedCitation" : "[4]", "plainTextFormattedCitation" : "[4]", "previouslyFormattedCitation" : "[4]" }, "properties" : { "noteIndex" : 0 }, "schema" : "" }[4] This Series (n=41)Fasano et al. (n=61)Mean Age (range) / Sex44(19-75), 81% F44 (19-66), 92% FMedian Duration (range)12 months (0-30)6.7 years (0-30)Side (R:L:Both*)40% R; 50% L; 10% B*31% R; 39% L; 29% B**Paroxsymal vs Persistent93% vs 7%60% vs 40%Location Eye Mouth (Down vs up) Unilateral Platysma Contraction/ Jaw Deviation Tongue deviation90%90% (90% vs 10%)85%12%17%51%84% (63% vs 26%)61%Not recorded84%Functional Movement Disorder Weakness Ipsilateral limb Contralateral limb Limb Dystonia78%71%7%22%18%Not recordedNot recordedArm (29%), Leg (16%), Neck (16%)Movement triggered by eye movement or sustained facial muscle contraction51%Not recordedOther Functional Symptoms Migraine Fatigue Convergence Spasm Dysphonia Non-epileptic attacks 22%12%22%12%32%26%18%Not recordedNot recordedNot recordedTriggering and premonitory symptoms17% Headache, 7% premonitory dissociationNot recorded*both eyes (n=1), episodic alternating lower face (n=3); ** subset of 51 patients with lip involvementReferences:ADDIN Mendeley Bibliography CSL_BIBLIOGRAPHY [1] Tan EK, Jankovic J. 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