Angioleiomioma oral mimetizando um granuloma piogênico: relato de caso raro / Oral Angioleiomyoma mimetizing a pyogenic granuloma: A rare case report

Angioleiomioma (AL) é um tumor benigno de origem perivascular que raramente é observado na cavidade oral, principalmente em localizações como a gengiva. Devido sua apresentação clínica inespecífica, os ALs podem mimetizar outras lesões orais, como tumores benignos de glândulas salivares e lesões reacionais, como o granuloma piogênico. O presente artigo objetiva relatar um caso raro de AL localizado em gengiva, em uma paciente de 19 anos. Clinicamente, a lesão apresentava-se como um tumor assintomático, oval, pedunculado, bem definido, com superfície lisa, consistência fibrosa e cor eritematosa, semelhante a um granuloma piogênico, Uma biópsia excisional foi realizada e o fragmento foi encaminhado para análise histopatológica, que revelou uma proliferação vascular de diversos calibres, contendo paredes musculares espessas e proliferação muscular adjacente, além de infiltrado inflamatório, predominantemente crônico, hemácias extravasadas e área de ulceração, consistente com o diagnóstico de AL inflamado. A histopatologia desempenha um papel importante no diagnóstico final de lesões raras e com características clínicas inespecíficas. A excisão cirúrgica da lesão é o tratamento de escolha mais eficaz para os ALs orais. (AU)

Angioleiomyoma (AL) is a benign tumor of smooth muscle of perivascular origin that is rarely seen in the oral cavity, mainly in locations like the gingiva. Due to their nonspecific clinical presentation, ALs can mimic other oral lesions, such as benign salivary gland tumors and reactional lesions, as a pyogenic granuloma. We reported a case of an AL located in the gingiva in a 19-year-old female patient. In clinical terms, the lesion was presented as an asymptomatic, oval, pedunculated, well-defined nodule with a smooth surface, fibrous consistency and erythematous color, similar to a pyogenic granuloma. An excisional biopsy was performed and the fragment was sent for histopathological analysis that revealed a vascular proliferation of different calibers, containing thick muscle walls and adjacent muscle proliferation, in addition to an inflammatory infiltrate, predominantly chronic, extravasated red blood cells and an area of ulceration, consistent with the diagnosis of inflamed AL. The histopathology plays an important role in the final diagnosis of rare lesions and with nonspecific clinical characteristics. The surgical excision of the lesion is the most effective treatment of choice for oral ALs. (AU)

Angioleiomioma en cavidad oral «una neoplasia rara en un sitio poco común¼. Reporte de un caso y revisión de la literatura / Angioleiomyoma in the oral cavity «a rare neoplasm in an unusual site¼. Case report and literature review

El angioleiomioma (AL) es una neoplasia benigna, bien circunscrita y de crecimiento lento, que representa 5% de las neoplasias de tejidos blandos y cuya etiología es desconocida. Se origina del músculo liso, mayormente de las paredes de los vasos sanguíneos; su localización es más frecuente en extremidades, siendo raros en la región de cabeza y cuello, y más aún en cavidad bucal. Histológicamente la lesión se caracteriza por ser un nódulo bien encapsulado con proliferación de fascículos de músculo liso maduro alrededor de la luz de los vasos sanguíneos, cuyas células suelen ser positivas a marcadores de inmunohistoquímica como alfa actina de músculo liso, desmina, HHF35, miosina, calponina y H-caldesmon. El tratamiento actual es la escisión quirúrgica completa con una tasa de recurrencia prácticamente nula. El objetivo es resaltar la importancia del diagnóstico y el manejo correcto de las lesiones intraorales a través de la presentación de un caso clínico de un leiomioma vascular localizado en región nasolabial, además de hacer la revisión de la literatura correspondiente (AU)

The angioleiomyoma (AL) is a benign neoplasm, well circumscribed and slow growing, that represents 5% of the soft tissue neoplasms, whose etiology is unknown. It originates from smooth muscle, mostly from the walls of blood vessels; regarding its location, it more frequently appears in the extremities, being rare in the head and neck region, and even more so in the oral cavity. Histologically, the lesion is characterized by being a well encapsulated nodule with proliferation of mature smooth muscle bundles around the lumen of the blood vessels, whose cells are usually positive for immunohistochemical markers such as alpha smooth muscle actin, desmin, HHF35, myosin, calponin and H-caldesmon. The current treatment is complete surgical excision having zero recurrence rate. The objective of the following article is to educate on the importance of correct diagnosis and management of intraoral lesions through the presentation of a clinical case of a vascular leiomyoma located in the nasolabial region, in addition to reviewing the corresponding literature (AU)

Angioleiomioma uterino: a propósito de un caso / Uterine angioleiomyoma: report of clinical case

Resumen El angioleiomioma es un tumor benigno perivascular que raramente se localiza en el útero. Se expone el caso de un angioleiomioma de gran tamaño en una mujer de 30 años con sangrado menstrual abundante y masa abdominal palpable. La paciente fue sometida a miomectomía y diagnosticada de angioleiomioma por el estudio histológico. Ante síntomas persistentes, la angiomiomectomía o la histerectomía simple son eficaces.

Abstract Angioleiomyoma is a benign perivascular tumor that is rarely located in the uterus. This paper presents a case of a large angioleiomyoma in a 30-year-old woman with heavy menstrual bleeding and a palpable abdominal mass. The patient underwent myomectomy and was diagnosed with angioleiomyoma by histological examination. For persistent symptoms, angiomyomectomy or simple hysterectomy are effective.

Leiomiomas cutáneos: revisión clinicopatológica y epidemiológica / Cutaneous leiomyomas: a clinicpathological and epidemiological review

Fundamento: Los leiomiomas cutáneos, superficiales o suprafasciales, se dividen en tres variantes: piloleiomiomas(PL), angioleiomiomas (AL) y leiomiomas genitales (LG) queincluyen las formas vulvares, escrotales y areolares. El objetivo fue establecer las características clinicohistológicas yla incidencia de cada variante, y las posibles asociacionescon neoplasias internas. Material y métodos: Se revisaron 255 casos de leiomiomascutáneos diagnosticados entre 1982 y 2018 en los serviciosde Anatomía Patológica de tres centros hospitalarios (Navarra y Alicante). Se describieron y compararon variables de-mográficas, clínicas, histológicas e inmunohistoquímicas. Resultados: La incidencia en Navarra de PL fue 4,3 casospor año y millón de habitantes, de AL 20 y de LG 1,4. Lasformas cutáneas suponen aproximadamente el 3,5 % deltotal de leiomiomas. La población con PL sufrió más frecuentemente cáncer de mama (OR = 4,8; IC95%: 1,3-17,4;p = 0,006). Los leiomiomas areolares son de pequeño tamaño, acompañados de dolor local, predominantementefasciculares o sólidos, con muy rara afectación del tejidocelular subcutáneo y escasa atipia. Esto contrasta con elresto de LG, de tamaño medio y rara vez dolorosos, predominantemente nodulares, con frecuente afectación deltejido celular subcutáneo y atipia. Conclusiones: Se aporta información sobre las características clinicohistológicas de las distintas variantes de leio-miomas, según las cuales debería replantearse la clasificación de los leiomiomas areolares fuera del grupo de LG. Sedetectó una asociación entre PL y carcinoma de mama quedeberá confirmarse en futuros estudios para determinar sieste leiomioma constituye un marcador de riesgo de cáncerde mama en mujeres.(AU)

Background: Cutaneous, superficial and or suprafascial leio-myoma are divided into three variants: piloleiomyomas (PL),angioleiomyomas (AL) and genital leiomyomas (GL) that include the vulvar, scrotal and areolar forms. This study setout to establish the clinical and histological characteristicsand incidence of each variant, and any likely associationswith internal neoplasms. Methods: A review was carried out of 255 cases of cutaneous leiomyomas diagnosed between 1982 and 2018 at thePathology departments of three hospitals (Navarra and Alicante). Demographic, clinical, histological and immunohis-tochemical variables were described and compared. Results: The incidence of PL in Navarra was 4.3 cases permillion inhabitants a year, with another 20 cases of AL and1.4 cases of GL. Cutaneous forms make up approximately3.5% of the total leiomyomas. The population with PL suffered more frequently from breast cancer (OR = 4.8; CI 95%:1.3-17.4; p = 0.006). Nipple leiomyomas are small, accompanied by localised pain, and are predominantly fascicularor solid, with very infrequent effect on the subcutaneouscellular tissue and scarce atypia. This makes for a contrastwith the other GLs, which are medium sized and infrequently painful, predominantly nodular, and frequent effect on thesubcutaneous tissue and atypia. Conclusions: The information provided here about the clinical and histological characteristics of the different varietiesof leiomyomas indicate that there is a need to reconsiderthe classification of nipple leiomyomas outside the group ofGLs. An association between PL and breast carcinoma wasdetected, which needs to be confirmed in future studies soas to determine if this leiomyoma is a risk marker for breastcancer.(AU)

[Renal angioleiomyoma with a myogenic and melanocytic immunophenotype, as a variant of angiomyolipoma]. / Angioleiomioma renal con patrón inmunohistoquímico miogénico y melanocítico como variante del angiomiolipoma.

We present a case of a 67 year old male with a cortical nodular tumour of the left kidney. During a year's follow-up with ultrasound and MRI, the tumour was seen to increase in size by 16-20 mm. The nodule was surgically removed. Microscopically it was well defined and unencapsulated, with a proliferation of typical fusiform cells of smooth muscle cell appearance, clumped around well vascularized areas. Immunohistochemically, the neoplasm was positive for muscle markers (smooth muscle actin, desmin and caldesmon) and melanocyte markers (HMB-45 and Melan A). Our case would appear to be a renal neoplasm with an angioleiomyomatous pattern, but with immunohistochemical characteristics of angiomyolipoma (PEComa), however, without either a lipomatous or lipid cell component. We found no previous reports of this type of tumour in the literature.

Angioleiomioma oral: relato de um caso e revisão dos achados atuais / Oral angioleiomyoma: case report and a review of current findings

Resumo O angioleiomioma é uma neoplasia benigna que, a partir da nova classificação da OMS (2013) para os tumores de tecidos moles, deixou de ser considerado um tumor de origem muscular lisa, passando a ser considerado um tumor de origem perivascular. Raramente os angioleiomiomas ocorrem na cavidade oral. A lesão é tratada cirurgicamente, com prognóstico considerado favorável. Este trabalho revisa os casos de angioleiomioma oral relatados na literatura nos últimos 5 anos e descreve esse tumor em um homem de 44 anos que apresentou um nódulo assintomático localizado em lábio superior, com evolução de 6 meses. As hipóteses diagnósticas foram de adenoma pleomórfico e adenoma canalicular. A lesão foi submetida à biópsia e análise histopatológica e imuno-histoquímica (S100, CD34, α-SMA, H-caldesmon e desmina) confirmaram o diagnóstico de angioleiomioma. Destacamos a imuno-histoquímica como um importante método auxiliar no diagnóstico diferencial do angioleiomioma com outras lesões e, principalmente, com o miopericitoma.

Abstract Angioleiomyoma is a benign neoplasm that was considered a tumor of smooth-muscle origin until the most recent (2013) WHO classification of soft tissue tumors, in which it was reclassified as a tumor of perivascular origin. Angioleiomyomas rarely occur in the oral cavity. These lesions are treated surgically with good prognosis. This article presents a review of reports of oral angioleiomyoma in the literature from the last 5 years and describes the case of a 44-year-old man who presented with an asymptomatic nodule in the upper lip that had developed over a 6-month period. Diagnostic hypotheses of pleomorphic adenoma or canalicular adenoma were raised. Biopsy of the lesion, histopathological and immunohistochemical analysis (S100, CD34, H-caldesmon, and desmin) confirmed a diagnosis of angioleiomyoma. It is noteworthy that immunohistochemistry is an important auxiliary method for differential diagnosis of angioleiomyoma from other tumors, particularly myopericytoma.

Angioleiomioma cavernoso cutáneo: Presentación de un caso / Cutaneous cavernous angioleiomyoma: A case report

El angioleiomioma es una neoplasia cutánea rara, benigna, que deriva de las células musculares lisas de los vasos sanguíneos. Se presenta habitualmente en los miembros inferiores de mujeres de edad mediana, como un tumor doloroso. El subtipo histológico cavernoso es poco frecuente. El tratamiento de elección es la extirpación quirúrgica de la lesión. Presentamos el caso de una paciente con un angioleiomioma cavernoso plantar izquierdo.(AU)

Angioleiomyoma is a rare, benign skin neoplasia derived from smooth muscle cells of blood vessels. It usually occurs in the lower limbs of middle-aged women as a painful tumor, palms and soles. The cavernous histological subtype is unusual. The treatment of choice is surgical removal of the lesion. We present a case of a patient with a left cavernous angioleiomyoma planting.(AU)

Angioleiomioma cavernoso cutáneo: Presentación de un caso / Cutaneous cavernous angioleiomyoma: A case report

El angioleiomioma es una neoplasia cutánea rara, benigna, que deriva de las células musculares lisas de los vasos sanguíneos. Se presenta habitualmente en los miembros inferiores de mujeres de edad mediana, como un tumor doloroso. El subtipo histológico cavernoso es poco frecuente. El tratamiento de elección es la extirpación quirúrgica de la lesión. Presentamos el caso de una paciente con un angioleiomioma cavernoso plantar izquierdo.

Angioleiomyoma is a rare, benign skin neoplasia derived from smooth muscle cells of blood vessels. It usually occurs in the lower limbs of middle-aged women as a painful tumor, palms and soles. The cavernous histological subtype is unusual. The treatment of choice is surgical removal of the lesion. We present a case of a patient with a left cavernous angioleiomyoma planting.

Oral angioleiomyoma: a case report and considerations on differential diagnosis / Angioleiomioma oral: relato de caso e considerações no diagnóstico diferencial

Angioleiomyomas are benign mesenchymal tumors derived from smooth muscle, which rarely occur in the oral cavity. We report a case of an angioleiomyoma occurring in the maxillary gingiva. The lesion was painless, with a lobulated surface, fibrous in consistency, and firm upon palpation. Microscopic examination showed an encapsulated tumor mass composed of large vascular channels of varying calibers, surrounded by thick walls of irregularly arranged, spindle-shaped cells, which were immunopositive for smooth-muscle actin. It is sometimes difficult to differentiate an angioleiomyoma from other spindle-cell tumors, thus we emphasize its histological differential diagnosis.

Angioleiomiomas são tumores mesenquimais benignos derivados de músculo liso que raramente ocorrem na cavidade oral. Relatamos um caso de angioleiomioma na gengiva maxilar. A lesão era indolor, com superfície lobulada, consistência fibrosa e firme à palpação. O exame histológico mostrou massa tumoral encapsulada, composta por grandes canais vasculares de diferentes calibres, circundados por paredes espessas de células fusiformes dispostas de forma irregular, as quais se apresentavam imunopositivas para actina de músculo liso. A distinção entre angioleiomioma e outros tumores de células fusiformes algumas vezes é difícil e, por essa razão, enfatizamos seu diagnóstico diferencial histológico.

Angioleiomiomas de la mano / Hand angioleiomyomas

Los angioleiomiomas son tumores benignos poco frecuentes en la mano. Tienen mayor incidencia a partir de la cuarta década de la vida, con igual distribución por sexo. Rara vez se realiza un diagnóstico preoperatorio correcto, pues suelen confundirse con gangliones y lipomas, entre otros. Se describen 11 casos clínicos tratados en el Centro de Diagnóstico Integral "Santa Bárbara de Zulia" en la República Bolivariana de Venezuela durante un trienio. Luego de la escisión simple la evolución fue favorable y no hubo recidiva.

Angioleiomyomas are rare benign tumors of the hand. They have higher incidence from the fourth decade of life, with equal gender distribution. A correct preoperative diagnosis is rarely made, because they are usually mistaken for ganglions and lypomas, among others. Eleven clinical cases treated at "Santa Bárbara de Zulia" Comprehensive Diagnosis Center in the Bolivarian Republic of Venezuela during a triennium are described. After the simple excision, outcome was favorable and there were not relapses.

Angioleiomiomas de la mano / Hand angioleiomyomas

Los angioleiomiomas son tumores benignos poco frecuentes en la mano. Tienen mayor incidencia a partir de la cuarta década de la vida, con igual distribución por sexo. Rara vez se realiza un diagnóstico preoperatorio correcto, pues suelen confundirse con gangliones y lipomas, entre otros. Se describen 11 casos clínicos tratados en el Centro de Diagnóstico Integral Santa Bárbara de Zulia en la República Bolivariana de Venezuela durante un trienio. Luego de la escisión simple la evolución fue favorable y no hubo recidiva(AU)

Angioleiomyomas are rare benign tumors of the hand. They have higher incidence from the fourth decade of life, with equal gender distribution. A correct preoperative diagnosis is rarely made, because they are usually mistaken for ganglions and lypomas, among others. Eleven clinical cases treated at Santa Bárbara de Zulia Comprehensive Diagnosis Center in the Bolivarian Republic of Venezuela during a triennium are described. After the simple excision, outcome was favorable and there were not relapses(AU)