Angioleiomyoma of the Sinonasal Tract: A Systematic Review of an Uncommon Clinicopathological Entity

Abstract Introduction Angioleiomyoma is a rare neoplasm that represents ∼ 0.2 % of all head and neck benign tumors and ∼ 2% of total cases of tumors of the sinonasal tract. It was once considered a possible subtype of leiomyoma, but, in the 2020 World Health Organization (WHO) classification of soft tissue tumors, it is accepted as a singular entity. Objective To systematically review the existing literature on angioleiomyoma in the light of the new classification of soft tissue tumors. Data Synthesis The present study was performed in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. A comprehensive search in the PubMed, Cochrane, Scopus, and Google Scholar databases was performed in January 2022. The search items included the following keywords: nasal angioleiomyoma OR sinonasal angioleiomyoma OR nasal vascular leiomyoma OR sinonasal vascular leiomyoma. A total of 87 patients were evaluated. He age of the patients in the studies ranged from 15 to 88 years (mean age at diagnosis: 55.6 years). The most common site of involvement was the nasal septum (28.4 %), followed by the inferior turbinate (22.5%). The most common symptom was nasal obstruction (66.7%), followed by epistaxis (47.1%). Surgical excision represented the main treatment, and there was recurrence of pathology in only 1 case (0.9%). Conclusion To our knowledge, only 87 cases of sinonasal-tract angioleiomyoma have been previously described. The results of the present review seem to confirm the rarity and the benign nature of this neoplasm, and they seem to confirm the necessity to improve the available data about sinonasal-tract angioleiomyoma.

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.

Leiomioma vascular que afecta el labio: reporte de un caso y un análisis actualizado de los 78 casos reportados en la literature / Vascular leiomyoma affecting the lip: report of a case and an updated analysis of the 78 cases reported in the literature / Leiomioma vascular que afeta o lábio: relato de um caso e análise atualizada dos 78 casos relatados na literatura

Resumen El leiomioma vascular (LV) es un tumor benigno de tejido blando que rara vez se observa en los tejidos orales. El objetivo de este trabajo es presentar un nuevo caso de leiomioma vascular del labio (VLL), describiendo sus características clínicas, microscópicas e inmunohistoquímicas, y revisar la literatura sobre el tumor. Una mujer de 27 años presentó una pequeña lesión dolorosa en la mucosa del labio inferior. La biopsia escisional reveló una masa encapsulada bien circunscrita formada por células pequeñas y fusiformes. El análisis de inmunohistoquímica reveló una expresión intensa y difusa de actina del músculo liso (AME) dentro de las células tumorales y la inmunorreactividad CD34 de las células endoteliales que recubren los espacios vasculares, lo que indica la presencia de vasos sanguíneos. Se estableció un diagnóstico final de VLL. No se observaron signos de recurrencia después de cinco años de seguimiento. Aunque VLL es una lesión rara, debe considerarse en el diagnóstico diferencial de las lesiones nodulares en los labios inferior y superior.

Abstract Vascular Leiomyoma (VL) is a benign soft tissue tumor rarely observed in oral tissues. The aim of this paper is to present a new case of vascular leiomyoma of the lip (VLL), describing its clinical, microscopical, and immunohistochemical features, and review the literature on the tumor. A 27-year-old woman presented with a small painful lesion on lower lip mucosa. Excisional biopsy revealed a well-circumscribed, encapsulated mass formed by small and fusiform cells. Immunohistochemistry analysis revealed intense and diffuse expression of smooth muscle actin (SMA) within the tumor cells and CD34 immunoreactivity of the endothelial cells lining the vascular spaces, indicating the presence of blood vessels. A final diagnosis of VLL was established. No signs of recurrence were observed after five years of follow-up. Although VLL is a rare lesion, it must be considered in the differential diagnosis of nodular lesions on lower and upper lips.

Resumo O Leiomioma Vascular é um tumor benigno de tecidos moles raramente observado nos tecidos da cavidade bucal. O objetivo desse artigo é apresentar um novo caso de um leiomioma vascular no lábio (LVL), descrevendo seus achados clínicos, microscópicos e imunoistoquímicos, e revisar a literatura sobre o tumor. Uma mulher de 27 anos de idade se apresentou com uma pequena lesão dolorosa na mucosa do lábio inferior. A biópsia excisional revelou um massa encapsulada e bem circunscrita, formada por células pequenas e fusiformes. A análise imunoistoquímica revelou expressão intensa e difusa de actina musculo liso (AML) nas células tumorais e imunorreatividade para CD34 em células endoteliais que revestem espaços vasculares, indicando a presença de vasos sanguíneos. O diagnóstico final de LVL foi estabelecido. Não foram observados sinais de recorrência após cinco anos de acompanhamento. Embora o LVL seja uma lesão rara, deve ser considerada no diagnóstico diferencial de lesões nodulares nos lábios inferior e superior.

Vascular Leiomyoma of the Nasal Floor: The Risk of Misdiagnosis

Angioleiomyoma of the oral cavity: a case report and brief review of the literature

This study presents a case of an oral angioleiomyoma along with its clinical diagnostic approach and laboratory confirmation. The differential diagnosis, especially from angioleiomyosarcoma, is also included. A 51-year-old patient presented with a tumor-like lesion on his upper labial mucosa. The clinical examination revealed a benign lesion that was surgically removed. Histopathological and immunohistochemical examinations confirmed the diagnosis of an oral angioleiomyoma. The post-surgical period was uneventful. No recurrence had occurred after a year of follow-up surveillance. Oral angioleiomyoma is a very rarely occurring oral lesion. Clinically, it may mimic some benign lesions, including fibroma, pyogenic granuloma or minor salivary gland tumor. Surgical excision is the treatment of choice. Histological and immunohistochemical examination can confirm the diagnosis. The differential diagnosis is crucial to rule out angioleiomyosarcoma.

Angioleiomyoma of the Nasal Cavity

Introduction Vascular leiomyoma of the nasal cavity is an extremely rare tumor that represents less than 1% of all vascular leiomyomas. It is more prevalent in women between the fourth and sixth decades, reaching primarily the inferior nasal turbinates. Objectives Reporting and assisting the systematization of more accurate diagnostic methods in clinical and complementary investigation of vascular leiomyoma in the nasal cavity. Resumed Report We present the case of a 49-year-old woman diagnosed with vascular leiomyoma in the nasal cavity, which manifested mainly with nasal obstruction. During investigation, computer tomography was not diagnostic, the cytologic study was not conclusive, and according to the biopsy, it was a squamous papilloma. Conclusion We suggest that the technical difficulty in obtaining an adequate amount of material for preoperative biopsy, associated with the topography of the lesion in the vestibular nasal region, may have contributed to changing the postoperative diagnosis. Thus, pathologic study of the surgical fragment is the more accurate method for diagnosis...

Vascular Leiomyoma in the Head and Neck Region: 11 Years Experience in One Institution

OBJECTIVES: Vascular leiomyoma is an uncommon benign tumor of smooth muscle origin that arises from the muscularis layer of blood vessel walls. We report our experiences with the clinical manifestations, pathologic characteristics, and management of vascular leiomyoma in the head and neck. METHODS: The clinical records of 12 patients with vascular leiomyoma of the head and neck in the 11-year period were reviewed retrospectively. RESULTS: The 12 patients included nine men and three women. The locations of the tumors were variable, including nasal cavity, auricle, hard palate, upper lip, upper eyelid, and supraclavicular space. All but three patients reported an asymptomatic spherical mass; the other three patients complained of intermittent epistaxis or unilateral nasal obstruction resulting from the tumor originating in the nasal cavity. All tumors were painless. Computed tomography consistently revealed a well-defined, intensely enhanced small mass on the mucosa. No case was dignosed corretly as vascular leiomyoma before surgical excision. All patients underwent localized surgical excision of the tumor without recurrence. Five of 12 tumors (42%) were of solid type, four (33%) were of venous type, and three (25%) were of cavernous in histological classification. The histologic type was not related to gender, site of occurrence, and presence of pain. CONCLUSION: Vascular leiomyoma presents as a small, painless mass in various locations of the head and neck region. Localized surgical excision is the only way to make the diagnosis and yields excellent results.

Color Doppler Ultrasonographic Findings of Vascular Leiomyoma: Pathologic Correlation

PURPOSE: To evaluate the distribution of color flow signals on color Doppler ultrasonography of vascular leiomyomas and to correlate them with pathologic findings. MATERIALS AND METHODS: We retrospectively analyzed color Doppler ultrasonographic images and pathologic slides of six vascular leiomyomas. We classified the patterns of distribution of color flow signals into localized compact cluster types and non-cluster types, and the pathologic findings into three subtypes: solid, venous and cavernous. RESULTS: All cases showed well-defined homogenous hypoechoic subcutaneous masses on gray-scale ultrasonography. Three cases showed localized compact cluster types on color Doppler ultrasonography, one in each subtype (solid, venous and cavernous). For the three non-cluster types, again there was on in each subtype. In addition, on pathologic analysis the zone of the localized compact cluster of color flow signals coincided with a cluster of larger, vascular caliber masses. CONCLUSIONS: Localized compact clusters of color flow signals on color Doppler ultrasonography were seen in 50% of our cases and correlated with a cluster of larger vascular caliber in the mass. But the pattern of distribution of color flows didn't show a correlation with pathologic type.

Angioleiomioma de cavidade nasal: relato de um caso e revisão de literatura / Nasal cavity vascular leiomyoma: case report and literature review

Leiomioma de cavidade nasal e seios paranasais é raro. Ele constitui menos de 1 por cento de todos os leiomiomas do corpo humano. Isto se deve à escassez de células musculares no nariz. Estas neoplasias podem ser classificadas em três grupos: leiomioma, angiomioma e leiomioma epitelióide. Somente 15 casos de angiomioma foram encontrados na literatura. O tratamento de escolha é a excisão cirúrgica. Um novo caso e a revisão da literatura são apresentados.

Liomyomas of the nasal cavity and paranasal sinuses are rare. They make up less than 1 percent of all leiomyomas in the human body. This is due to the paucity of smooth muscle in the nose. They are classified in three groups: leiomyoma, angiomyoma and epithelioid leiomyoma. Only 15 cases of vascular leiomyomas have been found in the literature. The treatment of choice is surgical excision. Hereby we present a new case and review the literature.

Angiomyoma of the Lingual Aspect of the Mandibular Second Molar: a Case Report

Angioleiomyoma of the Nasal Septum: A Case Report / 영남의대학술지

Angioleiomyoma of the sinonasal tract is a rare benign tumor. We report a case of angioleiomyoma of the nasal septum in a 51-year-old woman who complained of frequent epistaxis for 3 months. Surgicalexcision was performed. The excised specimen was 0.7 x 0.5 x 0.4 cm in size, well circumscribed, grayish white, rubbery, and soft. Histological examination showed thick-walled blood vessels and smooth muscle cell proliferation. No nuclear atypia or mitoses were present.

Leiomioma vascular nasal gigante: informe de un caso y su manejo endoscópico transnasal: revisión de la literatura / Giant nasal vascular leiomyoma: transnasal endoscopic management: report of a case and review of the literature

El leiomiona vascular intranasal es extremadamente raro. Menos de 1% de todos los leiomiomas se localizan en cabeza y cuello y de éstos sólo 3% se encuentran en la cavidad nasal. Los síntomas clínicos más comunes son obstrucción nasal, epistaxis y dolor. Se informa el caso de un leiomioma vascular nasal en una mujer de 35 años cuyo tratamiento se hizo mediante cirugía endoscópica transnasal para resecar el leiomioma vascular nasal más grande hasta el momento se comunica en la literatura.

An intranasal vascular leiomyoma is extremely rare. Less than 1% of all leiomyomas originate in the head and neck area, and of these, only 3% are found in the nasal cavity. The most common symptoms are nasal obstruction, epistaxis and pain. A rare case of nasal vascular leiomyoma that developed in a 35 year old woman is reported. Transnasal endoscopic sinus surgery to perform a resection on the biggest nasal vascular leiomyoma ever found was made.