ARTIGO ORIGINAL / ORIGINAL ARTICLE

 

Oral smooth muscle tumors: analysis of 49 cases and literature review

 

Tumores musculares orais: análise de 49 casos e revisão de literatura

 

 

Paula Prieto Oliveira^I; Fábio Daumas Nunes^I

^I Faculdade de Odontologia da Universidade de São Paulo (USP)- São Paulo- Brasil

Endereço para correspondência

 

 

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ABSTRACT

The aim of this study is to analyse and compare the clinical features of leiomyoma and leiomyosarcoma, because they help in the differential diagnosis. Records from Oral Pathology Department of Dental School at University of São Paulo during the period 1962-2010 were analysed. There were 49 cases of smooth muscle tumors: 27 leiomyomas and 22 leiomyosarcomas. Cases of smooth muscle tumors of the oral cavity were also compiled from 1970 to 2010. These tumors represented 0,092% of all oral lesions, with similar distribution between genders. Leiomyoma was more frequent in the fifth decade of life. The lips and the tongue were the most common sites. The majority of cases were asymptomatic. The accomplished treatment was surgical excision, with excellent prognosis. Leiomyosarcoma was more frequent in patients up to 50 years and the jaws were the most affected sites. The majority of cases were symptomatic and the pain was the most present symptom. Complete surgical resection was the most appropriate treatment. The recurrence rate was 27%, with 24% of metastasis and 30% of mortality, demonstrating the bad prognosis of this lesion.

KEYWORDS: Leiomyoma, leiomyosarcoma, epidemiology, mouth.

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RESUMO

O objetivo deste estudo foi avaliar e comparar as características clínicas de leiomioma e leiomiossarcoma de boca, visto que auxiliam no diagnóstico diferencial. Foram analisados 49 casos de tumores de músculo liso em cavidade oral diagnosticados no Departamento de Patologia Bucal da Faculdade de Odontologia da Universidade de São Paulo entre 1962 e 2010, sendo 27 leiomiomas e 22 leiomiossarcomas. Também foi realizado um levantamento de casos de tumores de músculo liso publicados na literatura entre 1970 e 2010. Foi observada uma frequência de 0,092% de tumores de músculo liso em boca, com distribuição semelhante entre os sexos. O leiomioma foi mais comum na quinta e sexta décadas de vida, sendo os lábios e a língua os locais mais afetados. A maioria dos casos eram assintomáticos. O tratamento realizado foi a excisão cirúrgica, com prognóstico excelente. Já o leiomiossarcoma apresentou maior frequência em pacientes acima de 50 anos, sendo os ossos gnáticos as regiões mais acometidas. A maioria dos pacientes eram sintomáticos e a dor foi o sintoma mais encontrado. O tratamento mais adequado foi a ressecção cirúrgica completa.O índice de recidiva foi de 27%, com 24% de metástases e 30% de mortalidade, demonstrando o prognóstico sombrio dessa lesão.

DESCRITORES: Leiomioma, leiomiossarcoma, epidemiologia, boca.

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Introduction

Smooth muscle tumors are very rare in the oral cavity due to the scarcity of smooth muscle in this region, and arise mainly from tunnica media of blood vessels. Leiomyomas represent from 0,016% to 0,064% of buccal benign lesions, while leiomyosarcomas are even less frequent^1,2,3,4.

The histologic distinction between atypical leiomyoma and low grade leiomyosarcoma is difficult and is based on the following criteria: number of mitosis, cellular atypia and pattern of necrosis^5,6. However, there isn't an exact number of mitosis or other features that warrants separation between both lesions. For this reason, clinical data are of paramount importance and must be considered in the diagnosis.

The aim of this retrospective study is to evaluate and compare clinical features, like gender, age, location and symptomatology of leiomyoma and leiomyosarcoma, and review the literature from 1970 to 2010, since there isn't any recent comparative article.

 

Material and Methods

Records of oral leiomyoma and leiomyosarcoma from Oral Pathology Department of Dental School at University of São Paulo during the period 1962-2010 were analysed. A review of published cases from 1970 to 2010 was accomplished by means of pubmed. Only english articles were considered. Data from both casuistry were analysed separately, including age, sex, race, location, symptomatology, treatment and prognosis. Cases with uncertain location were excluded.

 

Results

Nine thousand seven hundred and seventy histopathologic exams were accomplished during this period and only 64 (0,092%) were diagnosed as smooth muscle tumors, that corresponded to 49 cases, being 27 (55%) leiomyomas e 22 (45%) leiomyossarcomas. From 27 leiomyomas, 21 (78%) were vascular type and only 6 (22%) were solid, no epithelioid was found. The table 1 shows distribution of lesions according to gender. This datum was known in 47 cases. There was a male predominance in leiomyomas, with incidence of 61,5%, while leiomyosarcomas were more common in females, that corresponded to 57,1% of cases. The male-female ratio was 1,6:1,0 for leiomyoma and 1,0:1,3 for leiomyosarcoma.

 

 

 

The age was known in 43 cases (table 2). The leiomyoma ocurred mainly in the fifth decade of life, while leiomyosarcoma presented a more balanced distribution between age groups. The age of leiomyoma patients ranged from 8 to 78 years, with mean of 39 years; while age of leiomyosarcoma patients ranged from 1 to 70 years, with mean of 35 years.

 

 

 

For location (table 3), leiomyomas were more common in lips, tongue, jaws and alveolar ridge, while leiomyosarcomas were more frequent in the jaws, followed by alveolar ridge and cheeks.

 

 

 

There were 215 published cases of smooth muscle tumors from 1970 to 2010: 102 leiomyomas and 113 leiomyosarcomas. Out of 102 leiomyomas, 71 (69,6%) were vascular, 28 (27,4%) solid and 3 (3%) epithelioid. Table 4 shows distribution of lesions according to gender. This information was known in 99 cases. The men were more affected in leiomyomas, while leiomyosarcomas presented a similar distribution between genders. The ratio male-female was 1,2:1,0 for leiomyomas and 1,0:1,0 for leiomyosarcomas.

 

 

 

The age was known in 213 cases (table 5). Leiomyomas were more common in fifth and sixth decades of life, while leiomyosarcomas affected mainly the third decade of life. The age varied from 2 months to 85 years, with mean of 42 years, for leiomyoma, and from 6 to 97 years, with mean of 42,5 years for leiomyosarcoma.

 

 

 

For location (table 6), leiomyomas were more common in lips, hard palate and jaws, while leiomyosarcomas affected mainly the jaws, tongue, alveolar ridge and cheeks.

The symptomatology was related in 75 pacients with leiomyoma and 92 with leiomyosarcoma. Out of these cases, 75% and 40%, respectively, were asymptomatic. The main signs and symptons observed are described in table 7.

Surgical excision was accomplished for all leiomyomas with related treatment. For leiomyosarcomas, the main treatment of choice was surgical resection in the 102 patients with known treatment (table 8).

Follow up data were reported for 53 cases with leiomyoma and 95 cases with leiomyosarcoma. Only 3 (6%) leiomyomas were reported to recur. Out of leiomyosarcomas, 26 (27%) presented recurrence, 23 (24%) suffered metastases and 29 (30%) died of disease.

 

 

 

 

 

 

 

Discussion

The frequency of smooth muscle tumors in our study was very low and corresponded to 0,092% of all biopsies, as described in literature^1,4,7. There was a slight predominance of leiomyomas (55%) in relation to leiomyosarcomas (45%), although some authors report that only from 20% to 25% of oral smooth muscle tumors are malignant^1,8. The most common histological type of leiomyoma in both analyzed groups was vascular, as observed in others studies^{2,8,9,10,11,12,13,15,16}. However, Liang et al ¹⁷ found a slight predominance of solid leiomyoma.

Males were more affected in leiomyomas of both casuistics, as described by other articles^{1,2,7,14,15,16,17,18}. However, Brooks et al ¹⁹ and Gaitan-Cepeda et al ²⁰ found more females. Ravindranathan and Heslop²¹, Praal et al ²², Giles and Gosney²³, Yamamoto et al ²⁴ and Epivatianos et al ²⁵ didn't observed any gender predilection. For leiomyosarcomas, there was a slight predominance of women in our cases, while in literature cases no gender predilection was found. Dry et al ²⁶, Lo Muzio et al ²⁷, Vilos et al ³ and Centeno et al ²⁸ also observed a slightly higher incidence of females. However, many authors reported a slight predominance of men^{1,15,29,30,31,32,33,34,35}, and others observed intense predilection for males^{36,37,38,39,40,41}.

The age more affected for leiomyomas was the fifth decade of life in both groups, as described by others articles^{9,10,12,14,15,19,20,21,22,23,25,42,43}. However, Yamamoto et al ²⁴found more frequency in the third and fouth decades of life. For leiomyosarcomas, there was a similar distribution between the ages in our casuistic, while in the literature cases, there was a predominance of third decade, as reported for Vilos et al ³. However, the majority of articles observed higher incidence in older patients, up to 50 years^{15,27,30,33,35,41,44}. Yan et al ³⁴ found more frequency of second and fifth decades of life.

For location, the leiomyomas were more common in lips in both groups, as reported by others studies^{2,7,12,13,14,15,19,42,43,45}. However, some articles found higher incidence in tongue^{1,9,21,22,23,25}. The leiomyosarcomas affected mainly the jaws in both casuistics, agreeing with many studies^{3,15,26,29,30,33,34,39,40,46}. However, Muzio et al ²⁷ observed predominance of tongue and gums. Farman and Kay¹, Robinson and Whyte⁴⁷ found higher incidence in cheeks.

The majority of patients with leiomyoma were asymptomatic, as described in literature^{8,11,18,19,20,22,23,43,48,49,50}. However, according to Wertheimer-Hatch et al ¹⁵, only 40% were asymptomatic. For leiomyosarcomas, 60% of cases presented some kind of symptomatology and the most found symptom was pain, agreeing with other sutdies^15,26,30. However, according to Amarapala and Tilakaratne³², Ethunandan et al ³³ and Yan et al ³⁴, the most leiomyosarcomas are asymptomatic.

The treatment used for all leiomyomas was surgical excision and the prognosis was very good, with only 6% of recurrence, as described in literature^{2,8,15,16,17,18,19,21,22,45,50,51,52}. For leiomyosarcomas, the majority of cases suffered surgical resection, with 20% of recurrence and 15% of metastasis. For surgery followed by chemo/radiotherapy, there was 45% of recurrence and 20% of metastasis. Many authors question the eficiency of radio and chemotherapy in the control of these lesions and claim that complete surgical resection is the best treatment^{3,31,34,39,41,53,54,55,56,57,58}. However, Aydin and Dreyer⁵⁹ used radiotherapy in an inoperable leiomyosarcoma in posterior region of tongue and there was remission of the lesion, without evidence of recurrence and metastasis after one year and six months of follow up. Mitsudo et al ⁶⁰ accomplished chemotherapy with docetaxel and cisplatin using superselective intra-arterial infusion via superficial temporal artery for treatment of a leiomyosarcoma in the maxillary sinus and observed a reduction of more than 50% of tumor size. Abn et al ³⁵ accomplished chemotherapy with ifosfamide and doxorubicin in an incurable leiomyosarcoma of tongue, with multiple metastases, as a palliation and ocurred a significant reduction of the lesion.

In our study, there was recurrence in 27% of patients with leiomyosarcoma, 24% were affected by metastases and 30% died of disease, showing the bad prognosis of tumor. Some authors report even higher indices^15,30,32,33.

The literature review showed that smooth muscle tumors are very rare in the oral cavity and presented a similar distribution between genders. The leiomyoma is more common in the fifth decade of life and the most affected sites are lips and tongue. The majority of cases are asymptomatic. The treatment is surgical excision, with excelent prognosis and low recurrence index. The leiomyosarcoma presents higher incidence in people up to 50 years and affect mainly the jaws. The majority of patients are asymptomatic and the main symptom is pain. The best treatment is complete surgical resection. Radiotherapy is used in inoperable cases and chemotherapy as a palliation. The prognosis is bad, with high recurrence, metástase and mortality rates.

 

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Endereço para correspondência:
Av Dr Altino Arantes 620 ap 201 Vila Clementino
CEP: 04041-003 São Paulo-SP
Master's degree student Oral Pathology Department from Dental School at University of São Paulo (USP)
e-mail: paulaprietoterra@yahoo.com.br

 

Recebido para publicação: 05/11/12
Aceito para publicação: 28/01/13