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Leiomyosarcoma of the tongue: a case report
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British Journal of Oral and Maxillofacial Surgery 46 (2008) e69–e70
Short communication
Leiomyosarcoma of the tongue: a case report Tim Crossman ∗ , Paul Ward, J. Herold Maxillofacial Surgery BSUH Trust, United Kingdom Accepted 10 March 2008 Available online 29 April 2008
Abstract Primary leiomyosarcomas arising from within the tongue are extremely rare. Only eight cases have been previously reported on Medline to our knowledge. We describe a case of a 46-year-old woman who presented with a lesion rapidly increasing in size on the lateral aspect of her tongue, which was excised. We present a completed-5-year follow-up. © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Leiomyosarcoma; Tongue
Introduction Leiomyosarcoma is a malignant neoplasm that originates in smooth muscle. Visceral lesions are the most common, and account for about 7% of all soft-tissue sarcomas. The lesion is rare in the head and neck, making up 3%–10% of all leiomyosarcomas.1 Primary leiomyosarcoma of the tongue is extremely rare.2 To date we know of only eight cases reported of primary leiomyosarcoma of the tongue.2–4 This article describes an additional case of a primary oral leiomyosarcoma arising in the tongue. Clinical presentation A 46-year-old woman was referred by her general medical practitioner to the maxillofacial department with a lesion on the left side of her tongue, which had been present for four weeks and was rapidly increasing in size. Clinical examination showed a well-demarcated firm nodule that was painful to palpate. It was 1.5 cm in diameter and located in the body of the left side of her tongue. The overlying mucosa was indurated and ulcerated (Fig. 1). There were no palpable cervical lymph nodes and no signs or symptoms of a remote ∗
Corresponding author. E-mail addresses: [email protected] (T. Crossman), [email protected] (P. Ward), [email protected] (J. Herold).
primary tumour. We did an incisional biopsy under local anaesthetic. Histological examination showed an infiltrating spindle cell tumour that stained for smooth muscle actin. The presence of many abnormal mitoses and cytological atypia gave a diagnosis of leiomyosarcoma (Fig. 2). The chest radiograph was normal and magnetic resonance imaging indicated no disease in the neck. She was treated by hemiglossectomy. Access to the great vessels was gained through the neck, but we did no formal neck dissection. It was reconstructed with a radial forearm fasciocutaneous flap. The patient made a good functional and cosmetic recovery with no sign of recurrence, and was subsequently discharged from her five-year review.
Discussion Oral leiomyosarcoma is rare. Clinically, soft-tissue leiomyosarcoma often presents as a well-circumscribed, usually painless, mass that is firmly adherent to the surrounding soft tissues, which may have some degree of overlying ulceration.5 Histologically, tumour is locally aggressive and highly infiltrating,6 often leading to local recurrence and distant metastases.7 The cause of leiomyosarcoma remains uncertain, although cases associated with trauma, oestrogenic stimulation, ion-
0266-4356/$ – see front matter © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2008.03.008
e70
T. Crossman et al. / British Journal of Oral and Maxillofacial Surgery 46 (2008) e69–e70
Fig. 1. The lesion at presentation.
ment. Other publications suggest that the prognosis of oral leiomyosarcoma that affects the tongue is good if clear excision can be achieved. In the previous eight cases reported in which follow-up has ranged from one to five years there has been no evidence of recurrence.2 This suggests that primary leiomyosarcoma has a better prognosis if it arises within the tongue than elsewhere in the body. The recurrence rate for tumours that arise from, and are limited to, the dermis is reported to be between 14% and 42%.9 However, the case described is only the second case that we know of of primary leiomyosarcoma arising within the tongue, in which a five year follow-up has been completed before the case has been reported. The overall number of reported cases of this rare malignant lesion in the tongue remains extremely small, so prompt reporting of future cases and long term follow-up are required.
References
Fig. 2. Histopathological slide of the lesion (haematoxylin and eosin, original magnification ×352).
ising irradiation, and the Epstein-Barr virus have been reported.2 The treatment of choice for soft tissue lesions is excision with a margin of clear tissue,8 with supplemental dissection of regional lymph nodes, if there is evidence of nodal involve-
1. Barnes L, editor. Surgical pathology of the head and neck, Volume 1. New York: Marcel Decker; 1985. p. 814. 2. 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. 3. Mayall F, Hickman J, Bulman C, Blewitt R. Leiomyosarcoma of tongue: a very rare tumour. J Laryngol Otol 1994;108:617–8. 4. Goldberg MH, Polivy C, Saltzman S. Leiomyosarcoma of the tongue. J Oral Surg 1970;28:608–11. 5. Mesquita RA, Migliari DA, de Souza SO, Alves MR. Leiomyosarcoma of the buccal mucosa: a case report. J Oral Maxillofac Surg 1998;56:504–7. 6. Phelan JT, Sherer W, Mesa P. Malignant smooth muscle tumors (leiomyosarcomas) of soft tissue origin. N Engl J Med 1962;266:1027–30. 7. Freedman PD, Jones AC, Kerpel SM. Epithelioid leiomyosarcoma of the oral cavity: report of two cases and a review of the literature. J Oral Maxillofac Surg 1993;51:928–32. 8. Enzinger FM, Weiss SW, editors. Soft tissue tumors. 2nd ed. Mosby: St. Louis; 1988. p. 402. 9. Humphreys TR, Finkelstein DH, Lee JB. Superficial leiomyosarcoma treated with mohs micrographic surgery. Dermatol Surg 2004;30:108–12.
British Journal of Oral and Maxillofacial Surgery 46 (2008) e69–e70
Short communication
Leiomyosarcoma of the tongue: a case report Tim Crossman ∗ , Paul Ward, J. Herold Maxillofacial Surgery BSUH Trust, United Kingdom Accepted 10 March 2008 Available online 29 April 2008
Abstract Primary leiomyosarcomas arising from within the tongue are extremely rare. Only eight cases have been previously reported on Medline to our knowledge. We describe a case of a 46-year-old woman who presented with a lesion rapidly increasing in size on the lateral aspect of her tongue, which was excised. We present a completed-5-year follow-up. © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Leiomyosarcoma; Tongue
Introduction Leiomyosarcoma is a malignant neoplasm that originates in smooth muscle. Visceral lesions are the most common, and account for about 7% of all soft-tissue sarcomas. The lesion is rare in the head and neck, making up 3%–10% of all leiomyosarcomas.1 Primary leiomyosarcoma of the tongue is extremely rare.2 To date we know of only eight cases reported of primary leiomyosarcoma of the tongue.2–4 This article describes an additional case of a primary oral leiomyosarcoma arising in the tongue. Clinical presentation A 46-year-old woman was referred by her general medical practitioner to the maxillofacial department with a lesion on the left side of her tongue, which had been present for four weeks and was rapidly increasing in size. Clinical examination showed a well-demarcated firm nodule that was painful to palpate. It was 1.5 cm in diameter and located in the body of the left side of her tongue. The overlying mucosa was indurated and ulcerated (Fig. 1). There were no palpable cervical lymph nodes and no signs or symptoms of a remote ∗
Corresponding author. E-mail addresses: [email protected] (T. Crossman), [email protected] (P. Ward), [email protected] (J. Herold).
primary tumour. We did an incisional biopsy under local anaesthetic. Histological examination showed an infiltrating spindle cell tumour that stained for smooth muscle actin. The presence of many abnormal mitoses and cytological atypia gave a diagnosis of leiomyosarcoma (Fig. 2). The chest radiograph was normal and magnetic resonance imaging indicated no disease in the neck. She was treated by hemiglossectomy. Access to the great vessels was gained through the neck, but we did no formal neck dissection. It was reconstructed with a radial forearm fasciocutaneous flap. The patient made a good functional and cosmetic recovery with no sign of recurrence, and was subsequently discharged from her five-year review.
Discussion Oral leiomyosarcoma is rare. Clinically, soft-tissue leiomyosarcoma often presents as a well-circumscribed, usually painless, mass that is firmly adherent to the surrounding soft tissues, which may have some degree of overlying ulceration.5 Histologically, tumour is locally aggressive and highly infiltrating,6 often leading to local recurrence and distant metastases.7 The cause of leiomyosarcoma remains uncertain, although cases associated with trauma, oestrogenic stimulation, ion-
0266-4356/$ – see front matter © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2008.03.008
e70
T. Crossman et al. / British Journal of Oral and Maxillofacial Surgery 46 (2008) e69–e70
Fig. 1. The lesion at presentation.
ment. Other publications suggest that the prognosis of oral leiomyosarcoma that affects the tongue is good if clear excision can be achieved. In the previous eight cases reported in which follow-up has ranged from one to five years there has been no evidence of recurrence.2 This suggests that primary leiomyosarcoma has a better prognosis if it arises within the tongue than elsewhere in the body. The recurrence rate for tumours that arise from, and are limited to, the dermis is reported to be between 14% and 42%.9 However, the case described is only the second case that we know of of primary leiomyosarcoma arising within the tongue, in which a five year follow-up has been completed before the case has been reported. The overall number of reported cases of this rare malignant lesion in the tongue remains extremely small, so prompt reporting of future cases and long term follow-up are required.
References
Fig. 2. Histopathological slide of the lesion (haematoxylin and eosin, original magnification ×352).
ising irradiation, and the Epstein-Barr virus have been reported.2 The treatment of choice for soft tissue lesions is excision with a margin of clear tissue,8 with supplemental dissection of regional lymph nodes, if there is evidence of nodal involve-
1. Barnes L, editor. Surgical pathology of the head and neck, Volume 1. New York: Marcel Decker; 1985. p. 814. 2. 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. 3. Mayall F, Hickman J, Bulman C, Blewitt R. Leiomyosarcoma of tongue: a very rare tumour. J Laryngol Otol 1994;108:617–8. 4. Goldberg MH, Polivy C, Saltzman S. Leiomyosarcoma of the tongue. J Oral Surg 1970;28:608–11. 5. Mesquita RA, Migliari DA, de Souza SO, Alves MR. Leiomyosarcoma of the buccal mucosa: a case report. J Oral Maxillofac Surg 1998;56:504–7. 6. Phelan JT, Sherer W, Mesa P. Malignant smooth muscle tumors (leiomyosarcomas) of soft tissue origin. N Engl J Med 1962;266:1027–30. 7. Freedman PD, Jones AC, Kerpel SM. Epithelioid leiomyosarcoma of the oral cavity: report of two cases and a review of the literature. J Oral Maxillofac Surg 1993;51:928–32. 8. Enzinger FM, Weiss SW, editors. Soft tissue tumors. 2nd ed. Mosby: St. Louis; 1988. p. 402. 9. Humphreys TR, Finkelstein DH, Lee JB. Superficial leiomyosarcoma treated with mohs micrographic surgery. Dermatol Surg 2004;30:108–12.