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Schwannoma of the base of the Tongue - a case report of a rare disease and review of literatures.DR. Mohd. Yusuf Haider1, Dr. Md.Monjur Rahim2, Dr. N.M.K Bashar2, Dr. Md. Zakir Hossain3, Prof (Col) Dr. Sk Md Jaynul Islam31 Department of ENT, Colonel Malek Medical College, Manikganj2 Assistant Professor, ENT, Colonel Malek Medical College, Manikganj2 Dept. of ENT, Junior Consultant, 250 Bed District Hospital, Manikganj3 Asst. Professor, Dept. of Anesthesiology, Colonel Malek Medical College, Manikganj3 Histopathologist, Armed Forces Institute of Pathology, Dhaka Abstract: Around 25 – 40% of schwannoma happen within the head and neck locale, and of these, 1- 12% influence the intraoral range, most regularly the tongue or mouth floor. The point of this consider was to depict clinic pathologic and radiographic highlights of a case of schwannoma including the base of the tongue and to audit the writing of this abnormal clinical substance. Case reports with survey of the pathologic, radiologic and clinical information for patients with schwannoma of the tongue are detailed. Audit of the literary works of case reports of schwannomas (neurilemmomas) of the tongue from 1955 to 2019 with investigation of the patient’s age, sexual orientation, showing symptom(s), tumor measure, and surgical approach was embraced. The quiet in our thought displayed with effortless swelling of the tongue. Transoral extraction was performed and Histopathological and immunohistochemical examination affirmed the conclusion of schwannoma. A add up to of 129 cases of schwannoma of the tongue have been detailed within the English writing over the past 63 a long time. Schwannomas of the tongue regularly display within the third decade of life (33%), show no sex inclination (52.8% female; 47.2% male) and frequently display as an easy mass (69.6%). Schwannomas are likely to inspire upsetting indications when they happen within the back one-third of the tongue (63.2 vs. 13.5%) or approach 3 cm in most prominent measurement (33.0 vs. 18.2 mm). The tremendous lion's share of cases have been treated with transoral extraction (94.8%). Recurrence after surgical extraction has not been detailed. Schwannoma of the tongue could be a generally uncommon tumor of the head and neck. Transoral resection permits for expulsion of this tumor in a way that blocks repeat, maintains a strategic distance from causing dreariness of tongue work, and remains the standard approach for the treatment of the tremendous lion's share of these tumors.Keywords: Schwannoma, Neurilemmoma, Lingual, Base of the Tongue.Introduction:322834043053000147637542767250Also known as neurilemmoma, Schwannoma is a benign nerve sheath tumor. It was first identified by Virchow in 1908. These tumors can emerge from any nerve covered with a Schwann cell sheath, including the cranial nerves (with the exception of the optic and olfactory nerves), the spinal nerves, and the autonomous nervous system [1]. When the nerve of origin is small, it can be difficult to demonstrate its connection with a given tumor. On the other hand, if the site of origin is a larger nerve, it is observed that the nerve fibers are splayed over the outer side of the capsule instead of being absorbed into the tumor mass [2]. Virchow defined it for the first time in 1908. About 25–45% of all schwannomas occur in the head and neck CITATION Eun17 \l 1033 (Eun-Young Lee*, 2017)[3]. Around 1–12 million of these occur intraorally [4, 5] with the language being the most common site [5, 6]. Although there are many case reports of tongue schwannomas in the literature, after Hatziotis et al. [6] there has been no comprehensive review of the literature. We present two cases of tongue schwannoma and study the literature available from the last 51 years (1955–2006). Materials and methods A PubMed search for the terms "tongue schwannoma," "lingual schwannoma," "tongue neurilemmoma," and "lingualneurilemmoma" was conducted with the 1955–2006 date range. The search was restricted to English case reports. Unless the ventral tongue was also involved, mouth floor Schwannomas were not included. All the case reports had confirmed the masses ' identity as schwannomas histologically. From the case reports for data analysis, the following elements were extracted: age, gender, location of schwannoma (anterior one-third vs. posterior two-thirds of tongue), symptoms, tumor size, and treatment modality.Figure-1: Large asymptomatic swelling at posterolateral aspect of right side of tongue. 411480062484023907755975356667505880103390900906145457527889635Figure-2: Axial T1 & T2 and coronal T2 weighted Magnetic Resonance Image showing a well-defined mass centered right to the midline in the posterior base of the tongue. The tumor has a smooth well- defined border, with no invasion to the adjacent muscle.Figure-3: Fragment-of-spindle-cells.Case Report:We report a case of 28 year old male farmer, with a swelling at the posterior aspect of the right side of tongue. One year back this swelling was accidentally discovered by his newly married wife, while he was yawning. He was absolutely asymptomatic except occasional feeling of stickiness in the throat. The patient had no difficulty in chewing, swallowing or phonation and also no sensory or taste abnormalities. With the instance interest of the wife, the patient went to the capital city and got admitted into a government hospital. After fifteen days, the impatience patient and his ardent wife left the hospital stealthily with the lame excuse of delay. After that, the patient got admitted into another renowned hospital, a very good surgical team was preparing the patient for the operation but without any reason he left the hospital secretly. Finally the young, eager and worried couple came to Manikganj 250 bedded district hospital at the department of ENT to me and I found it as a rare case and shown my keen interest for academic purpose.3181350316865010572753191510On examination there was an oval swelling at the right side of base of the tongue measuring about 4cm X 3cm. The surface was smooth, margin regular, no discoloration or distortion of tongue epithelium. It was farm in consistency and was not fixed with underlying or overlying structures. The remaining oral cavity examination was normal, with no cervical lymph node enlargement. My first clinical diagnosis was dermoid cyst or Lipoma. He was evaluated with FNAC which revealed benign mesenchymal spindle cell neoplasm, favour, nerve sheath tumor possibly schwannoma. MRI of the tongue manifested hyper intense well circumscribed soft tissue mass in right half of base of the tongue. The patient underwent trans-oral total excision of the mass under general anesthesia with nasotracheal intubation. For the proper visualization of the base of tongue, frenulum of tongue was incised; tongue was released from floor of mouth and pulled out. An incision was given in right lateral margin over the swelling. After splitting the mucosa, mass is exposed, mobilized by blunt dissection and excised totally. Homeostasis is secured and wound closed in layers. Histopathological report revealed features of Schwannoma. For confirmation of the tissue of origin immunocytochemistry was done and found strongly positive for S100 protein.ba3152775351155847725372745Fig-4: Tumor seen on the base of the tongue187579035242500Fig-5: a) encapsulated mass, b) tumor removed from the tongue.Fig-6: Excised mass from the base of the tongue.B areaA area1238250235585Fig-7: The lesion is composed of spindled cells with hepercellular Antoni A areas and hypocellular Antoni B areasDiscussion:Though this is not clear of the etiology of the schwannoma, it is known to be derived from nerve sheath Schwann cells, which surround cranial, peripheral, and autonomic nerves [6, 7]. The head and neck are rather common location of this neoplasm. Intraoral Schwannoma mainly arise from the tongue, followed by the palate, mouth floor, buccal mucosa, gingiva, lip, and vestibule [8, 9], though the tongue is most commonly involved [10]. The lesion is slow growing, and thus, its onset is usually long before presentation. Lingual schwannoma shows no age or gender predisposition [11]. Usually, it is presented as a painless lump in any part of the tongue of average size2.4 cm. However, when the mass exceeds 3.0 cm, dysphagia, pain (or discomfort), dysphonia, and voice change are usually presented (Table 1).In this 60 years period from 1959 to 2019 we have noticed that, the total number of patients identified with Schwannomas around 68 and 54% of them are male and rest of them are female patients around 45%. More than half of the total patients have suffered in posterior Schwannomas (56%) and according to this information, the patients had lump, pain and dysphagia accordingly 54%, 3% and 22%. The mean age at diagnosis was nearly 25 years, standard treatment was transoral excision, performed in 96% cases. However, for tumors located at the base of the tongue, in two cases Carbon dioxide laser used and in three cases submandibular approach had taken. There were no reports of recurrence. Congenital lesions can be subcategorized into vascular and nonvascular categories (Fig-8) CITATION Jac17 \l 1033 (Jack E. Steiner, 2017). Lesions of the root of the tongueInfectiousNeoplasticCongenitalNon Vascular- lingual thyroid- Thyroglossal duct cyst- Epidermoid cyst- Lipoma-HemangiomaVascular- Lymphatic malformation- Venous malformation- venolymphatic malformation-Artcriovenous malformationMalignant*Rhabdomyosarcoma*Alveolar soft part carcomaBenign*Rhabdomyoma*Schwannoma*LeomyomaAbscessClinically, the schwannomas may be indistinguishable from other encapsulated benign tumors, so that biopsy and histological examination are essential to formulate a correct diagnosis. An excisional biopsy was performed to formulate a correct diagnosis and finally find out that the case was uncomplicated. In the present case before done the surgery of the patient he was totally asymptomatic and had not any injury after surgery. The option of complete resection was chosen on the basis of lesion form and size and to avoid recurrence. Table-1: Patients and tumor characteristics of tongue schwannomasAuthorYearGenderAgeSize (cm)SitePresentationSurgical approachMercantini and Mopper [21]1959M221AnteriorIntermittent painTransoralCameron [22]1959M251.5AnteriorLumpTransoralChadwick [23]1964F202.2PosteriorLumpTransoralCraig [24]1964F83PosteriorLumpTransoralPantazopoulos [25]1965F454.5PosteriorDysphasia/change in voiceTransoralChamber [26]1965M295PosteriorThroat discomfortTransoralFifer et al. [27]1966F283AnteriorLumpTransoralHatziotis and Aspride [28]1967M25HazelnutPosteriorLumpTransoralOles and Werthemier [29]1967M521AnteriorLumpTransoralPaliwal et al. [30]1967M322.5AnteriorLumpTransoralCrawford et al. [31]1968M230.5AnteriorLumpTransoral1968M241AnteriorLumpTransoralDas Gupta et al. [32]1969F215PosteriorPainTransoralBitici [33]1969M402.5AnteriorSlight discomfortTransoralSinha and Samuel [34]1971M231.5PosteriorDysphagiaTransoralMosadomi [35]1975M193AnteriorPainful massTransoralSwangsilpa et al. [36]1976M263AnteriorLumpTransoralSharan and Akhtar [37]1978F301.5AnteriorChange in voiceTransoralAkimoto et al. [38]1987M151AnteriorLumpTransoralSira et al. [39]1988F183PosteriorLumpTransoralFlickinger et al. [40]1989F283AnteriorLumpTransoralTalmi et al. [41]1991F751PosteriorLumpTransoralGallesio and Berrone [42]1992F211.9Anterior/baseDysphonia/paresthesia/chewing difficultyTransoralLopez and Ballistin [10]1993M240.6AnteriorLumpTransoralHaring [43]1994F492AnteriorLumpTransoralNakayama et al. [44]1996F405.5AnteriorLumpTransoralDreher et al. [15]1997F313BaseDysphagiaTransoralSpandow et al. [45]1999M377.9PosteriorThroat discomfortTransoralde Bree et al. [2]2000F245Posterolateral/baseLumpSubmandibularPfeifle et al. [46]2001F300.3AnteriorLumpTransoralCinar et al. [47]2004M71AnteriorLumpTransoralBassichis and McMlay [48]2004M92.3Posterior/baseSnoringTransoralNakasato et al. [49]2005F92Posterolateral/baseBleeding/ulcerationTransoralHwang et al. [50]2005M232.8AnteriorLumpTransoralLopez-Jornet and Bermejo-Fenoll [51]2005M390.8Posterolateral/baseLumpTransoralVafiadis et al. [52]2005M183.1AnteriorLumpTransoralBansal et al. [53]2005M264Posterolateral/ventralParesthesia/dysphoniaTransoralHsu et al. [7]2006M205Posterior/baseBleedingTransoralYing et al. [54]2006F264Posterior/baseDysphagia/otalgiaTransoralEnoz et al. [14]2006M72.5Anterior/baseDysphagia/painTransoralMehrzad et al. [55]2006M492.2Posterior/ventralPainCO2-transoralBatra et al. [56]2007M303Posterolateral/baseDysphagia, dyspnea, abscessTransoralBallesteros et al. [57]2007F312BasePainCO2-transoralSawhney et al. [19]2008F374.6Posterolateral/baseDysphagia/snoringSubmandibularSethi et al. [58]2008F281Anterolateral/ventralLumpTransoralPereira et al. [59]2008M121.5Posterolateral/ventralLumpCohen and Wang [17]2009M770.7Posterolateral/ventralLumpTransoralGupta et al. [60]2009F181Anterior/ventralLumpTransoralMardanpour and Rahbar [61]2009M182PosteriorDysphagia/change of voiceTransoralKaraca et al. [62]2010F132Posterolateral/ventralDysphagiaTransoralCigdem et al. [63]2010M132Anterior/ventralLumpTransoralJeffcoat et al. [64]2010M681.5LateralLumpTransoralNaidu and Sinha [65]2010M122Anterolateral/baseParesthesia/bleeding/ulcerationTransoralLuk?i? et al. [66]2011M101.5Posterolateral/ventralLumpTransoralBatra et al. [67]2011F384.2Posterior/ventralDysphagia/change of voiceTransoralNisa et al. [68]2011F388.5Posterolateral/ventralDysphagia/dysphonia/dyspneaTransoralMonga et al. [69]2013M202Posterolateral/baseLumpTransoralLira et al. [5]2013F262.5Posterior/ventralCervical painTransoralErkul et al. [70]2013M213Posterolateral/ventralChewing difficultyTransoralJayaraman et al. [71]2013F253Anterolateral/baseLumpTransoralGeorge et al. [4]2014M264Posterolateral/baseDysphagia/dysphoniaTransoralBhola et al. [11]2014F141.5Anterolateral/ventralLumpTransoralMoreno-García et al. [16]2014F132Anterior/ventralLumpLip split/mandibulotomyNibhoria et al. [72]2015F181.5Posterolateral/ventralLumpTransoralGopalakrishnan et al. [73]2016M323Posterolateral/ventralDysphagiaTransoralSharma and Rai [74]2016F204Posterolateral/ventralDysphagia/dysphoniaTransoralKav?i? and Bo?i? [75]2016F201.3Anterolateral/ventral/tipLumpTransoralLee et al. [76]2016M284Posterior/ventralLumpTransoralZain et al. 2016F24Not clearPosteriorlumpTransoralSteffi Sharma et al. 2018F204PosteriorlumpTransoralCurrent2019M284posteriorLumpTransoralConclusions: The authors report a report a case of lingual Schwannoma over the base of the tongue with preoperative FNAC and MRI. The mass was completely removed intraorally. No sign or symptoms of recurrence were observed at 12 months postoperatively. Despite the rarity of this condition, physicians should consider Schwannoma as a differential diagnosis for a mass over the tongue, as there can be a favorable outcome and prognosis for the patient when this condition is correctly identified. The definitive diagnosis requires a histopathological evaluation. Treatment is complete surgical excision of the lesion which does not result in any recurrence. Conflict of Interests:The authors declare that there is no conflict of interests regarding the publication of this paper.References:1. Harada H, Omura K, Maeda A. A massive pleomorphic adenoma of the submandibular salivary gland accompanied by neurilemomas of the neck misdiagnosed as a malignant tumor: report of case. J Oral Maxillofac Surg. 2001; 59:931–935.2. de Bree R, Westerveld G, Smeele L. Submandibular approach for excision of a large schwannoma in the base of the tongue. Eur Arch Otorhinolaryngol.2000; 257:283–286.3. Nelson W, Chuprevich T, Galbraith D. Enlarging tongue mass. 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