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Inicio Revista Española de Cirugía Oral y Maxilofacial Leiomyosarcoma of the tongue: A rare case report
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Vol. 39. Núm. 4.
Páginas 191-252 (Octubre - Diciembre 2017)
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Vol. 39. Núm. 4.
Páginas 191-252 (Octubre - Diciembre 2017)
Clinical report
DOI: 10.1016/j.maxilo.2016.10.004
Open Access
Leiomyosarcoma of the tongue: A rare case report
Leiomiosarcoma de la lengua: un caso clínico raro
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Carlos Alberto Salcedo-Gil
Autor para correspondencia
salcedocarlosmd@hotmail.com

Corresponding author.
, Víctor Alejandro Lasa-Menéndez, María Josefa Pastor-Fortea, José Ignacio Iriarte-Ortabe
Department of Oral and Maxillofacial Surgery, Son Espases University Hospital, Carretera Valldemossa 79, Planta 0 módulo Q. Sala 004, 07120 Palma de Mallorca, Spain
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Table 1. Reported cases of primary leiomyosarcoma arising from the tongue.
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Introduction

Leiomyosarcomas is rare tumour that arises from smooth muscle cells.1,2 They account for 7% of soft tissue sarcomas.1–3 Leiomyosarcoma in the head and neck region account for only 3–10%,2,4 and less than 0.1% develops in the oral cavity.2 It has been reported that oral leiomyosarcoma shows a bimodal peak in the third, sixth and seventh decades of life.4 Although a possible association with trauma, estrogenic stimulation, ionizing radiation and Epstein–Barr virus has been suggested, its underlying aetiology remains uncertain.4,5,9,10 This article describes an additional case of oral leiomyosarcoma affecting the tongue related to ionizing radiation.

Case presentation

A 41-year-old male patient who two years previously had undergone radiotherapy and chemotherapy because of a Cavum Carcinoma, the patient was referred to the maxillofacial department on October of 2014 because a rapidly increasing lesion on the on the right side of the tongue was noticed at the follow-up. Clinical examination showed a well-circumscribed firm, submucosal mass on the right side of the tongue (Fig. 1). No cervical lymphadenopathy was palpable. As control of his Cavum Carcinoma the patient had a PET/CT scan that only reports a focal uptake on right side of the tongue.

Fig. 1.

Clinical photograph showing the mass within the right side of the tongue.

(0,29MB).

An incisional biopsy was repeated on January 2015 after a first negative result; the histopathology showed an infiltrating spindle cell tumour that stained for smooth muscle actin. The immunohistochemistry stained positively with smooth muscle actin, desmin, and caldesmon (Fig. 2).

Fig. 2.

Microscopic examination showing interlacing fascicles of spindle-shaped cells (haematoxylin–eosin stain, original magnification, ×400).

(0,41MB).

He was treated with hemiglossectomy. It was reconstructed with a medial sural perforator flap, and no neck dissection was performed on March of 2015 (Fig. 3a–c). The patient had a good functional and cosmetic result with no signs of recurrence in subsequent follow-ups, last one on July 2016.

Fig. 3.

(a) Medial sural perforator artery, (b) immediate post-operative reconstruction and (c) postoperative follow up, good cosmetic outcome with a medial sural perforator flap and no recurrence.

(0,44MB).
Review of literature

The cases reported in the literature are summarized in Table 1. We included Oral LMS that only affected the tongue; publish in the English and Spanish literature with all the variables included in the table. The exclusion criteria were LMS not affecting the tongue and publication with incomplete medical reports.

Table 1.

Reported cases of primary leiomyosarcoma arising from the tongue.

Study  Year  Gender  Age (yrs)  Site  Metastasis  Treatment  Follow-up (yrs) 
Yannopoulos and Stout  1962  11mo  Tip of tongue  No  Excision  4–6 
O’Day et al.  1964  Sublingual  Yes  Excision  2; deceased 
O’Day et al.  1964  19  Sublingual  Yes  Excision and neck dissection  11 
Goldberg et al.  1970  54  Sublingual  No  Excision  1.5 
Brandjor d et al.  1977  63  Floor of the mouth  Yes  Excision 
Mindell et al.  1975  59  Tongue - Not specified  No  Excision 
Lack  1986  2.5  Base of tongue  No  Excision and chemotherapy 
Aydin and Dreyer  1994  70  Base and dorsum of tongue  No  Radiotherapy  1.5 
Mentzel et al.  1994  88  Floor of the mouth  Yes  Excision  10d, decease 
Mayall et al.  1994  60  Tip of tongue  No  Excision 
Piattelli and Artese  1995  80  Lateral border of tongue  No  Patient rejected treatment  N/A 
Crossman et al.  2008  46  Lateral border of tongue  No  Excision 
Sarra et al.  2009  62  Lateral border of tongue  Yes  No treatment  6 mo 
Yan et al.  2010  48  Floor of mouth  No  Excision and suprahyoid neck dissection  1.5, deceased 
Pires et al.  2010  55  Lateral border of tongue  No  Excision 
Ahn et al.  2011  54  Lateral border of tongue  Yes  Palliative chemotherapy  1mo 
Toranzo et al.  2012  73  Lateral border of tongue  No  Excision 
Shütz  2013  25  Floor of the mouth  No  Excision and neck dissection  6.5 
Present case  2016  41  Lateral border of tongue  No  Excision  1.5 
Discussion

Intraoral leiomyosarcoma, particularly in the tongue are extreme rare. They arise from smooth muscle cells.1,2,5–11 Intraoral leiomyosarcoma occurs more frequently in males.4 Clinically the tumour presents as a well-circumscribed painless mass. The correct diagnosis is only made following a definitive histological examination and confirmed by immunohistochemistry.3,6,8,10,11 Immunohistochemical staining has consistently shown positive for desmine, vimentin, and smooth muscle actine, whereas a negative response to S100 protein and the cytokeratins is usually found.1–7

Leiomyosarcoma behaves as an aggressive tumour, and has a tendency to infiltrate surrounding tissues. The more frequent distant metastases of leiomyosarcoma are the head and neck region,2–5 in 15% of the cases.2,3 Metastases can also spread to the regional lymph nodes, and the lung and the liver are prediction sites for distant metastases.2–5

The cause of leiomyosarcoma remains unknown, although cases associated with trauma, estrogenic stimulation, ionizing radiation and Epstein–Barr virus have been suggested.4,5,9,10

The treatment consists of wide surgical excision with regional lymph node dissection as required, with the goal of histologically clear margins. This determines the true course of the disease and the long-term prognosis.2–5,7–11

Chemotherapy is generally reserved for palliative cases, such as inoperable tumours or metastatic spread.2,8,10

The case that we report of a primary intraoral leiomyosarcoma it could be induced by ionizing radiation. It is essential that the clinician be familiar with the lesion, and anticipates the possible presence of this disease and makes early diagnosis. Treatment of primary leiomyosarcoma is radical tumour resection with histologically clear margins.

Ethical disclosuresProtection of human and animal subjects

The authors state that no experiments were performed on human beings or animals as part of this investigation.

Confidentiality of data

Authors state they have followed the protocols of their workplace about the data publication of patients.

Right to privacy and informed consent

Authors have obtained the informed consent from the patients or subjects referred to in the article. This document is in possession of the corresponding author.

Conflict of interest

The authors declare that there are no conflicts of interest.

References
[1]
G.A. Vilos, A.D. Rapidis, G.D. Lagogiannis, C. Apostolidis.
Leiomyosarcomas of the oral tissues: clinicopathologic analysis of 50 cases.
J Oral Maxillofac Surg, 63 (2005), pp. 1461-1477
[2]
A. Schütz, R. Smeets, O. Driemel, S.G. Hakim, H. Kosmehl, H. Hanken, et al.
Primary and secondary Leiomyosarcoma of the oral and perioral region – clinicopathological and immunohistochemical analysis of a rare entity with a review of the literature.
J Oral Maxillofac Surg, 71 (2013), pp. 1132-1142
[3]
M. Ethunandan, C. Stokes, B. Higgins, A. Spedding, C. Way, P. Brennan.
Primary oral leiomyosarcoma: a clinico-pathologic study and analysis of prognostic factors.
Int J Oral Maxillofac Surg, 36 (2007), pp. 409-416
[4]
J.H. Anh, T. Mirza, P. Ameerally.
Leiomyosarcoma of the tongue with multiple metastases: a case report and review of literature.
J Oral Maxillofac Surg, 70 (2012), pp. 1745-1750
[5]
S.-W. Yang, T.-M. Chen, C.-Y. Tsai, C.-Y. Lin.
A peculiar site of leiomyosarcoma: the tongue tip—report of a case.
Int J Oral Maxillofac Surg, 35 (2006), pp. 469-471
[6]
K. Sakamoto, K. Matsuzaka, M. Yama, T. Kakizawa, T. Inoue.
A case of leiomyosarcoma arising from the tongue.
Oral Oncol Extra, 41 (2005), pp. 49-52
[7]
A. Ketabchi, N. Kalavrezos, L. Newman.
Sarcomas of the head and neck: a 10-year retrospective of 25 patients to evaluate treatment modalities, function and survival.
Br J Oral Maxillofac Surg, 49 (2011), pp. 116-120
[8]
H. Amarapala, W.M. Tilakaratne.
Leiomyosarcoma of the oral cavity: report of seven cases and review of literature.
Oral Oncol Extra, 42 (2006), pp. 14-17
[9]
T. Crossman, P. Ward, J. Herold.
Leiomyosarcoma of the tongue: a case report.
Br J Oral Maxillofac Surg, 46 (2008), pp. e69-e70
[10]
J.M. Toranzo-Fernández, R. Colunga, L.G. González-Valdez.
Leiomiosarcoma del borde lateral de la lengua: reporte de un caso.
Rev Esp Cir Maxilofac, 36 (2014), pp. 27-31
[11]
L. Lo Muzio, G. Favia, M.D. Mignogna, A. Piattelli, E. Maiorano.
Primary intraoral leiomyosarcoma of the tongue: an immunohistochemical study and review of the literature.
Oral Oncol, 36 (2000), pp. 519-524
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