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   Table of Contents      
Year : 2016  |  Volume : 2  |  Issue : 2  |  Page : 82-84

Leiomyosarcoma of tongue: A case report and review of literature

Department of Radiotherapy, Sanjay Gandhi Post Graduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India

Date of Web Publication2-Jan-2017

Correspondence Address:
Mranalini Verma
Department of Radiotherapy, Sanjay Gandhi Post Graduate Institute of Medical Sciences, Lucknow - 226 014, Uttar Pradesh
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/2454-6798.197376

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Leiomyosarcoma (LMS) of the tongue is an extremely rare mesenchymal tumor. Till now, we came across about 24 cases of tongue LMS reported in the literature. Here, we are presenting the case of a 50-year-old female with 4 months history of ulcerative growth on the tongue along with difficulty in swallowing and tongue movement who was diagnosed with LMS of the tongue on histopathology. He was managed with surgical excision followed by radiotherapy without any recurrence or metastasis after 6 months of follow-up.

Keywords: Carcinoma; leiomyosarcoma; tongue

How to cite this article:
Verma M, Lal P. Leiomyosarcoma of tongue: A case report and review of literature. Asian J Oncol 2016;2:82-4

How to cite this URL:
Verma M, Lal P. Leiomyosarcoma of tongue: A case report and review of literature. Asian J Oncol [serial online] 2016 [cited 2018 May 21];2:82-4. Available from: http://www.asjo.in/text.asp?2016/2/2/82/197376

  Introduction Top

Leiomyosarcoma (LMS) is malignant neoplasm originating from smooth muscle account for 3-7% of soft tissue sarcomas which occurs frequently in uterine myometrium, gastrointestinal tract, retroperitoneum, skin, and subcutaneous tissue, [1] but rare in the oral cavity because of paucity of smooth muscle in that site, but when present they are usually localized on the tongue, lips, and palate. [2] It may arise as primary, radiation-associated, or metastatic tumor. [3] Primary LMS of the tongue is an exceedingly rare. Here, we describe a case of LMS of the tongue in a 50-year-old female.

  Case Report Top

We present the case of a 50-year-old female with an LMS of the tongue. She had reported to Oncosurgery Department with the complaint of ulcerative growth on the tongue along with difficulty in closing mouth and tongue movement for the last 4 months. She was tobacco-chewer for last 20 years with no other addiction history. She denies any exposure to chemicals, drugs, and sharp tooth. Her family history was also not significant. Her vital parameters were within normal range that she was conscious, alert, with blood pressure of 120/70 mm Hg, respiratory rate of 20/min, pulse rate of 110/min, and oxygen saturation of 92% on room air and her systemic evaluation was unremarkable. On local examination, she had a large ulcerative growth (size ~ 5 cm × 5 cm) arising from the right anterolateral aspect of anterior tongue. Computed tomography scan imaging showed heterogeneously enhancing mass of size 4.5 cm × 3.5 cm × 5.4 cm on right anterolateral aspect of anterior tongue along with loss of fat interface between the mass and underlying intrinsic muscles of tongue [Figure 1]. Biopsy of mass lesion revealed a malignant epithelial neoplasm and immunohistochemistry (positive for alpha smooth muscle actin and desmin, and negative for cytokeratin and S-100) are suggestive of LMS. Then, she was hospitalized and underwent wide excision right side glossectomy with Type II modified radical neck dissection. The definitive histopathological diagnosis performed on surgical specimen revealed LMS of tongue with absence of neoplastic infiltration of surgical resection margin and all 14 lymph nodes; results of immunohistochemical study were positive for alpha-smooth muscle actin and desmin, negative for cytokeratin and S-100 protein further re-enforces the same diagnosis.
Figure 1: Compute tomography scan showing heterogeneously enhancing mass of size 4.5 cm × 3.5 cm × 5.4 cm on right anterolateral aspect of anterior tongue along with loss of fat interface between mass and underlying intrinsic muscles of tongue

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In view of tumor depth of 3 cm and tumor size more than 5 cm, patient was for postoperative radiotherapy with a dose of 60 Gy/30 fractions/6 weeks with X-6MV on a linear accelerator without concomitant chemotherapy.

  Discussion Top

LMS is an uncommon malignant mesenchymal neoplasm originating from smooth muscle. It occurs frequently in the gastrointestinal tract and female genital tract. [1] Due to paucity of smooth muscle in head and neck region, only 3-7% of LMS cases occur in head and neck region. [1] However, when present in head and neck region, they are usually localized on the tongue, lips, and palate. [2] The cause of LMS is still uncertain, although association with trauma, estrogen therapy, ionizing radiation, and Epstein-Barr virus has been documented in the literature. [4]

Clinically, LMS often presents as a rapidly growing, painless, discrete mass firmly adherent to surrounding soft tissue. [4] LMS become large by the time diagnosis is confirmed because of very few associated symptoms. [4] Due to nonspecific clinical presentation, diagnosis of LMS is based primarily on pathologic criteria. Immunohistochemistry or electron microscopy must be carried out to achieve more specific differential diagnosis. [5],[6] The histological criteria include the presence of pleomorphism, bizarre cell forms, pattern of interlacing bundles of smooth muscle cells, and high mitotic rate. [7] Immunohistochemical study of LMS was consistently positive for alpha smooth muscle actin, vimentin, desmin, and negative for S-100 protein and cytokeratins. [8],[9]

The only effective treatment is complete resection with sufficient tumor-free borders and postoperative radiotherapy when necessary. [10] Radical neck dissection is needed in cases with lymphadenopathy. However, there is insufficient evidence to support the efficacy of radiotherapy and chemotherapy. Till now, only one case was reported who was treated with radiotherapy alone because of surgically inoperable condition. [9] In this case, no local recurrence or distant metastasis was reported after 1.5 years of follow-up. In another two cases, radiotherapy at a dosage of 65 Gy/28 fractions/3 months was used after surgical excision. [9],[11] Chemotherapy was recommended in cases with inoperable and metastatic disease. [12] A combination of ifosfamide and doxorubicin was used in one case. [7] However, further research is needed to document the efficacy of adjuvant therapy in LMS of tongue. Prognosis of tongue LMS is good if clear surgical margin is achieved after excision.

Till now, we only know of 34 cases reported (including our patient's case) of primary LMS of the tongue with four cases from India [11],[13],[14] including one of the authors [Table 1]. Review of 34 cases revealed that there were 18 males and 16 females with an age range from 1 year to 97 years with no predilection for any specific age group. The sites of tumor lesions in the tongue include the tip, the lateral border, and the base. In 93% (24/26) patients, excision is the main treatment.
Table 1: Previously reported cases of primary leiomyosarcoma of tongue

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This case report highlights that although LMS of the tongue is a rare mesenchymal tumor, we should be familiar with this unusual lesions because early diagnosis and aggressive management are the mainstay of therapy.

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Conflicts of interest

There are no conflicts of interest.[35]

  References Top

Croce A, Moretti A, Laus M, Crescenzi D. Leiomyosarcoma of the base of the tongue and free edge of the epiglottis: A case report. J Med Case Rep 2012;6:400.  Back to cited text no. 1
Luaces Rey R, Lorenzo Franco F, Gómez Oliveira G, Patiño Seijas B, Guitián D, López-Cedrún Cembranos JL. Oral leiomyoma in retromolar trigone. A case report. Med Oral Patol Oral Cir Bucal 2007;12:E53-5.  Back to cited text no. 2
Azevedo RS, Pires FR, Gouvêa AF, Lopes MA, Jorge J. Leiomyosarcomas of the oral cavity: Report of a radiation-associated and a metastatic case. Oral Maxillofac Surg 2012;16:227-32.  Back to cited text no. 3
Schenberg ME, Slootweg PJ, Koole R. Leiomyosarcomas of the oral cavity. Report of four cases and review of the literature. J Craniomaxillofac Surg 1993;21:342-7.  Back to cited text no. 4
Hashimoto H, Daimaru Y, Tsuneyoshi M, Enjoji M. Leiomyosarcoma of the external soft tissues. A clinicopathologic, immunohistochemical, and electron microscopic study. Cancer 1986;57:2077-88.  Back to cited text no. 5
Sonobe H, Furihata M, Hayashi K, Takahashi K, Ohtsuki Y, Kishimoto S. Poorly differentiated leiomyosarcoma of the maxillary sinus: A histological, immunohistochemical and ultra-structural study. J Clin Electron Microsc 1987;20:219-27.  Back to cited text no. 6
Ahn JH, Mirza T, Ameerally P. Leiomyosarcoma of the tongue with multiple metastases: A case report and review of literature. J Oral Maxillofac Surg 2012;70:1745-50.  Back to cited text no. 7
Piatteli A, Areste L. Leiomyosarcoma of the tongue: A case report. J Oral Maxillofac Surg 1995;53:M698-701.  Back to cited text no. 8
Aydin H, Dreyer T. Leiomyosarcoma of the base of the tongue treated with radiotherapy: A case report. Eur J Cancer B Oral Oncol 1994;30B:351-5.  Back to cited text no. 9
Ethunandan M, Stokes C, Higgins B, Spedding A, Way C, Brennan P. Primary oral leiomyosarcoma: A clinico-pathologic study and analysis of prognostic factors. Int J Oral Maxillofac Surg 2007;36:409-16.  Back to cited text no. 10
Dhanasekaran SV, Nair JS, Joyce ME. Leiomyosarcoma of tongue. Chrismed J Health Res 2014;1:271-3.  Back to cited text no. 11
  Medknow Journal  
Wertheimer-Hatch L, Hatch GF 3 rd , HatchB SK, Davis GB, Blanchard DK, Foster RS Jr, et al. Tumors of the oral cavity and pharynx. World J Surg 2000;24:395-400.  Back to cited text no. 12
Tandon DA, Fernandes P, Maheswari A, Tickoo SK. Leiomyosarcoma of the base of tongue. Indian J Otolaryngol Head Neck Surg 1996;48:235-7.  Back to cited text no. 13
Jain A, Singh SN, Singhal P,Sharma MP, Samadni S. Primary leiomyosarcoma of tongue: A rare neoplasm. J Evol Med Dent Sci 2014;3:473-8.  Back to cited text no. 14
Blanc E. Travaux originaux. Gaz Hebd Med Chir 1884;21:611.  Back to cited text no. 15
Gorsky M, Epstein JB. Head and neck and intra-oral soft tissue sarcomas. Oral Oncol 1998;34:292-6.  Back to cited text no. 16
Stout AP. Leiomyoma of the oral cavity. Am J Cancer 1938;34:31.  Back to cited text no. 17
Burford W, Ackerman L, Robinson H. Leiomyoma of the tongue. Am J Orthod Oral Surg 1944;30:395.  Back to cited text no. 18
Yannopoulos K, Stout AP. Smooth muscle tumors in children. Cancer 1962;15:958-71.  Back to cited text no. 19
de Bertelli AP. Uncommon tumors of the tongue. Oral Surg 1965;19:771.  Back to cited text no. 20
MacDonald DG. Smooth muscle tumours of the mouth. Br J Oral Surg 1969;6:207-14.  Back to cited text no. 21
Goldberg MH, Polivy C, Saltzman S. Leiomyosarcoma of the tongue: Report of case. J Oral Surg 1970;28:608-11.  Back to cited text no. 22
Lack EE. Leiomyosarcomas in childhood: A clinical and pathologic study of 10 cases. Pediatr Pathol 1986;6:181-97.  Back to cited text no. 23
Wollenberg B, Müller-Höcker J, Wustrow TP. Leiomyosarcoma of the tongue. Laryngorhinootologie 1993;72:342-5.  Back to cited text no. 24
Mayall F, Hickman J, Bulman C, Blewitt R. Leiomyosarcoma of the tongue: A very rare tumour. J Laryngol Otol 1994;108:617-8.  Back to cited text no. 25
Dry SM, Jorgensen JL, Fletcher CD. Leiomyosarcomas of the oral cavity: An unusual topographic subset easily mistaken for nonmesenchymal tumours. Histopathology 2000;36:210-20.  Back to cited text no. 26
Lo Muzio L, Favia G, Mignogna MD, Piattelli A, Maiorano E. Primary intraoral leiomyosarcoma of the tongue: An immunohistochemical study and review of the literature. Oral Oncol 2000;36:519-24.  Back to cited text no. 27
Vora NM, Levin RJ. Metastatic leiomyosarcoma to the tongue. Otolaryngol Head Neck Surg 2003;128:601-2.  Back to cited text no. 28
Sakamoto K, Matsuzaka K, Yama M, Kakizawa T, Inoue T. A case of leiomyosarcoma arising from the tongue. Oral Oncol Extra 2005;41:49-52.  Back to cited text no. 29
Kazemian A, Kamian SH, Hoseini MS, Azizi MR. Leiomyosarcoma of the tongue: Report of a case. Iran J Radiat Res 2005;3:143-7.  Back to cited text no. 30
Yang TL, Chiang CP, Kok SH, Kuo YS. Leiomyosarcoma of the tongue: A case report. Clin J Oral Maxillofac Surg 2006;17:109-16.  Back to cited text no. 31
Yang SW, Chen TM, Tsai CY, Lin CY. A peculiar site of leiomyosarcoma: The tongue tip - Report of a case. Int J Oral Maxillofac Surg 2006;35:469-71.  Back to cited text no. 32
Castaldi A, Arcuri T, Carta M, Quilici P, Derchi LE. Primary leiomyosarcoma of the oral tongue: Magnetic resonance and ultrasonography findings with histopathologic correlation. Acta Radiol 2006;47:514-7.  Back to cited text no. 33
Crossman T, Ward P, Herold J. Leiomyosarcoma of the tongue: A case report. Br J Oral Maxillofac Surg 2008;46:e69-70.  Back to cited text no. 34
Pires CA, Pires LF, Faber PA. A primary leiomyosarcoma of the lateral border of the tongue. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:e31-3.  Back to cited text no. 35


  [Figure 1]

  [Table 1]


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