RESUMO

The microscopical diagnosis of radicular cyst (RC) is straightforward; nonetheless, in some cases with unusual histopathological features, strict clinicopathological correlation is necessary to achieve the correct diagnosis. We report a case of a 5-year-old girl was referred presenting extensive carious lesion in the tooth #55, associated with vestibular sinus tract. Medical history revealed allergic asthma diagnosis. After clinical and imagi-nological exams, the deciduous tooth was extracted. Microscopically, the soft tissue lesion attached to the root showed typical RC features; however, in the cystic capsule, lymphoid follicles and vascular networks (lined by epithelioid endothelial cells) surrounded by nu-merous eosinophils, were observed. Immunohistochemistry, through CD3, CD20, CD34 and alpha-smooth muscle actin antibodies, highlighted these findings. Moreover, CD1a and CD207 were negative. To the best of our knowledge, this is the first report of RC showing angiolymphoid hyperplasia with eosinophilia (ALHE)-like features. (AU)

RESUMO

Background: Kimura's disease is an infrequent inflammatory disorder, of unknown etiology, with few reports outside of Asia. It presents as a nodule or tumor predominantly in the postauricular region, neck and parotid gland. It is histologically characterized by follicular hyperplasia with wellformed mantle zones, preservation of nodal architecture, prominent eosinophilic infiltrate in the germinal centers and interfollicular areas; and associated with elevated levels of IgE and peripheral eosinophilia. Clinical case: We present a case of a 23-year-old man from Mexico, he presented with a recurrent tumor in the right parotid gland, previously treated with surgical resection. Imaging studies were performed and a primary neoplasm of the salivary gland was suspected, he was treated with surgical resection. The histological diagnosis was Kimura's disease. Conclusions: Communication and divulgation of this rare inflammatory disorder expans the knowledge for the differential diagnosis of tumors of the head and neck, and salivary glands, mainly in men with peripheral eosinophilia and elevated IgE; it can sometimes simulate malignant neoplasms, leads to inadequate diagnostic and therapeutic approaches.

Introducción: la enfermedad de Kimura es un desorden inflamatorio poco frecuente, de etiología desconocida y raramente reportado fuera del continente asiático. Se presenta como un nódulo o tumor predominantemente en la región retroauricular, cervical o glándula parótida. Se caracteriza histológicamente por hiperplasia folicular con zonas del manto bien formadas, preservación de la arquitectura ganglionar, infiltrado eosinofílico prominente en los centros germinales y áreas interfoliculares; generalmente asociada a niveles elevados de IgE y eosinofilia periférica. Caso clínico: presentamos el caso de un hombre de 23 años, de origen mexicano que se presentó con un tumor recidivante a dos años de resección quirúrgica previa en glándula parótida derecha, se realizaron estudios de imagen y se sospechó de neoplasia primaria de glándula salival, fue tratado con resección quirúrgica. El diagnóstico histológico fue de enfermedad de Kimura. Conclusiones: la comunicación y difusión de este raro desorden inflamatorio amplía la base del conocimiento para el diagnóstico diferencial de tumores de cabeza y cuello, y glándulas salivales, predominantemente en hombres con eosinofilia periférica y elevación de IgE; que en ocasiones puede simular neoplasias malignas, llevando a abordajes diagnósticos y terapéuticos inadecuados.

RESUMO

Abstract Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare inflammatory disease of unknown origin, which may be asymptomatic or produce symptoms like pruritus, pain, or spontaneous bleeding, and which has no sex predilection. The treatment options are cryosurgery, pulsed dye laser, topical tacrolimus, surgery, curettage, and oral retinoids. Below is a report of a clinical case of angiolymphoid hyperplasia with eosinophilia in the oral cavity, as well as the treatment performed. This was a female patient who attended a stomatology and oral surgery clinic due to a localized lesion at the level of the vestibule between dental organs 11 and 12. Clinically, there was a nodular lesion of about 1 cm in diameter, located at the level of attached gingiva; it was asymptomatic on palpation and of unknown etiology. Angiolymphoid hyperplasia is an unusual pathology in the oral cavity; its etiology is unknown, and a definitive diagnosis should be made through histopathological examination and clinical signs.

Resumen La hiperplasia angiolinfoide con eosinofilia (HACE) es una enfermedad rara de origen desconocido, de carácter inflamatorio, que puede ser asintomática o presentarse con prurito, dolor o sangrado espontáneo, sin predilección por género. Las opciones de tratamiento son la criocirugía, el láser de colorante pulsado, el tacrolimus tópico, la cirugía, el curetaje y los retinoides orales. Se reporta un caso clínico de hyperplasia angiolinfoide con eosinofilia en cavidad oral, así como el tratamiento realizado. Paciente de sexo femenino, que acudió a consulta de estomatología y cirugía oral por presentar lesión localizada a nivel de vestíbulo entre órganos dentarios 11 y 12. Clínicamente se observó lesión nodular localizada en encía adherida de aproximadamente 1 cm de diámetro, asintomático a la palpación y de etiología desconocida. La hyperplasia angiolinfoide es una patología inusual en cavidad oral, de etiología desconocida, en la que el diagnóstico definitivo debe hacerse a través del examen histopatológico y los signos clínicos.

RESUMO

La hiperplasia angiolinfoide con eosinofilia es una enfermedad vascular o tumor vascular, infrecuente, tumoral o reactiva, con carácter inflamatorio, benigna y de etiología desconocida. Los hallazgos histológicos consisten en la proliferación vascular de células endoteliales prominentes y un infiltrado inflamatorio intersticial crónico de leucocitos, histiocitos y eosinófilos. Afecta frecuentemente a mujeres de la tercera y cuarta década de la vida. Se han descrito múltiples variantes de tratamiento pero es frecuente su recurrencia. Clínicamente se presenta como pápulas o nódulos subcutáneos, limitados, únicos o múltiples, color rojo, marrón o violáceo, que se localizan con frecuencia a nivel de la cabeza, cuello; en ocasiones se asocia a prurito y dolor. El interés en presentar este caso radica en su rara localización, su presentación en el sexo masculino; el tamaño, la extensión y la infrecuencia de la lesión. Se presenta el caso de un paciente masculino de 42 años de edad con múltiples lesiones en el eje longitudinal del miembro superior izquierdo, la mayor de 15 x 15 cm, localizada en la cara póstero-interna del brazo en sus dos tercios dístales, limitada, móvil, no dolorosa, con desplazamiento del tejido muscular, y deformidad estructural, con diagnóstico, después de unabiopsia posquirúrgica,de hiperplasia angiolinfoide con eosinofilia según histología La evolución del paciente fue satisfactoria y se mantiene seguimiento médico para comprobarposibles recidivas(AU)

The angiolymphoid hyperplasia with eosinophilia is a vascular disease (or vascular tumor) uncommon, tumoral or reactive, with inflammatory characteristics, benign of unknown etiologic. The histological finds consist on the vascular proliferation of endotelial prominent cells and a chronic inflammatory interstitial infiltrator of leukocytes, histiocytes and eosinophils. It frequently affect to elder women in the third and fourth ages. Clinically, it is presented as papules or subcutaneous nodules, limited, only one or multiple nodules; in red, brown or purplish color that are frequently located at the level of the head, neck; in occasions, it is associated with pruritus and pain. The interest in presenting this case is due to its rare localization, its appearance in the male sex, the size, extension and infrequency of the lesion. The case consists in a 42 year-old masculine patient with multiple lesions in the left superior member's longitudinal axis. The biggest lesion is of 15 x 15 cm, located in the postero-internal part of the arm in its distals two thirds, limited, movable, not painful, with displacement of the muscular tissue, and structural deformity, with a diagnostic of angiolymphoid hyperplasia with eosinophilia according to histology (post-surgical biopsy).The evolution of the patient was satisfactory, and remains medically followed-up in case of possible relapses(AU)

RESUMO

Abstract Angiolymphoid hyperplasia with eosinophilia is a rare and benign vascular tumor whose etiology remains uncertain. It clinically presents itself by angiomatous papules or nodules located on the head and neck. Many controversies in the literature are found in relation to angiolymphoid hyperplasia with eosinophilia and Kimura's disease - its main differential diagnosis - due to their clinical and histopathological similarities. However, currently, most studies agree that they are distinct diseases. The present case illustrates a characteristic description of angiolymphoid hyperplasia with eosinophilia and also highlights the main differences with Kimura's disease.

RESUMO

Angiolymphoid hyperplasia with eosinophilia is a rare disease considered as being a vascular malformation resulting from a subjacent arteriovenous shunt. It affects mostly the head of women between 20 and 40 years old and may present spontaneous involution.

RESUMO

The epithelioid hemangioma, also called angiolymphoid hyperplasia with eosinophilia, is a rare benign vascular tumor that manifests as nodules. Histopathological examination shows vascular spaces of various sizes lined by prominent endothelium, and inflammatory infiltrates composed of eosinophils, histiocytes, mast cells and lymphocytes. We report a case of epithelioid hemangioma characterized by skin lesion in the left upper limb. This is an unusual location, since the lesions occur most often in the head and neck.

O hemangioma epitelioide, também denominado hiperplasia angiolinfoide com eosinofilia, é um tumor vascular raro, de caráter benigno, que se manifesta como nódulos. O exame histopatológico evidencia espaços vasculares de diversos calibres, revestidos por endotélio proeminente e infiltrado inflamatório composto de eosinófilos, histiócitos, mastócitos e linfócitos. Relatamos um caso de hemangioma epitelioide caracterizado por lesão de pele no membro superior esquerdo. Essa é uma localização incomum, uma vez que as lesões ocorrem mais frequentemente na cabeça e no pescoço.

RESUMO

La expansión tisular es una técnica que aprovecha las propiedades viscoelásticas de la piel para distenderla mediante infiltraciones seriadas a través de una válvula de un dispositivo de silicona. dicho tejido se utiliza posteriormente para reconstrucción y para cubrir defectos. La hiperplasia angiolinfoide con eosinofilia es una enfermedad vasoproliferativa que se manifiesta clínicamente por tumores nodulares. se presenta el caso clínico de un paciente con expansión tisular, y en quien se encuentra de forma incidental, en la histopatología de la cápsula, hiperplasia angiolinfoide con eosinofilia. para el conocimiento del grupo a cargo del presente trabajo, este es el primer caso en el cual ambas entidades se relacionan. © 2012 Instituto Nacional de Cancerología. publicado por elsevier españa, s.L. todos los derechos reservados.

Tissue expansion is a technique that takes advantage of the viscoelastic properties of the skin by distending it using a series of injections through a valve of a silicone device. this tissue is subsequently used for repair and to cover defects. angiolymphoid hyperplasia with eosinophilia is a vasoproliferative disease that manifests clinically as nodular tumors. the clinical case is presented of a patient with tissue expansion in whom angiolymphoid hyperplasia with eosinophilia was an incidental finding in the histopathology of the capsule. To the knowledge of the group presenting this work, this is the first in which both conditions are linked.

RESUMO

Descreve-se um paciente com características clínicas e histopatológicas típicas da hiperplasia angiolinfoide com eosinofilia. Trata-se de uma hiperplasia vascular benigna que importa reconhecer e distinguir da doença de Kimura.

This report describes a patient with clinical and histopathological findings compatible with angiolymphoid hyperplasia with eosinophilia. This benign vascular hyperplasia should be recognized and differentiated from Kimura's disease.

RESUMO

Relata-se um caso clássico de hemangioma epitelioide encontrado em localização incomum - região vulvar - em uma paciente de 62 anos. Abordam-se aspectos histopatológicos, além do comportamento biológico e prognóstico da lesão.

This is a report of a 62 years old patient with haemangioma epithelioid in a very uncommon site. We study histopathological aspects, biological behaviour and prognosis.

RESUMO

A hiperplasia angiolinfoide (Hale e doença de Kimura são duas entidades que podem se manifestar como nódulos, placas ou pápulas, de localização predominante em face, região retroauricular e cervical. São causas raras de eosinofilia e ainda há muita discussão em torno de suas etiopatogenias. Para alguns autores trata-se de duas patologias distintas enquanto para outros são manifestações diferentes da mesma doença. O presente artigo relata o caso de um paciente asiático que apresentava história de prurido generalizado há um ano, acompanhado de pápulas em membros e nódulo de aproximadamente 5 cm de diâmetro em região retroauricular direita com aumento progressivo. O hemograma apresentava leucocitose às custas de eosinofilia. Os achados sugerem uma superposição entre as duas patologias, pois clinicamente são sugestivos de doença de Kimura, mas a histopatologia e imuno-histoquímica confirmaram a origem endotelial da lesão, sendo compatível com Hale. Os autores destacam a raridade do caso como causa de eosinofilia, assim como alertam para a dificuldade do diagnóstico e da diferenciação entre as duas patologias.

Angiolymphoid hyperplasia (Hale) and Kimura disease are two entities that can manifest as subcutaneous nodules, plaques, or papules, and the predominant localization is face, retroauricular and cervical region. They are rare causes of eosinophilia and there is discussion regarding their etiopathogenesis. Some authors propose that these two diseases are truly individual, but others say that they are separate points on a single spectrum. This article is a case report of an asian patient that showed a one year duration of generalized pruritus, with papules on the limbs and a 5 centimeter nodule on the right retroauricular region with slow progression of the size. The hemogram showed leucocitosis with eosinophilia. These findings suggest an overlap between these two diseases, they are clinically suggestive of Kimura disease diagnosis, but the histopathology with HE and immunohistochemistry confirmed the endothelial origin of the lesion that was compatible with Hale. The authors emphasize the rarity of the case as the cause of eosinophilia, also alert for the challenge of the diagnosis and distinction between these two diseases.

RESUMO

CONTEXT: Epithelioid hemangioma or angiolymphoid hyperplasia with eosinophilia is an uncommon benign vascular neoplasm that is usually located on the face or neck. Exceptionally, it has been described affecting the colon, with only two such cases described in the worldwide literature. The aim here was to present a case of primary epithelioid hemangioma of the sigmoid colon with confirmation by immunohistochemical examination. CASE REPORT: A 37-year-old woman had had a complaint of intermittent abdominal pain for six months. Two months after the condition started, she began to present changes in her intestinal habit, with evacuations containing blood and mucus and a weight loss of 4 kg over this period. At physical examination, a palpable mass was noted in the lower left quadrant of the abdomen. Neoplasia of the colon was clinically suspected and she underwent colonoscopy. This demonstrated the presence of a vegetating sessile lesion of approximately 5 cm in diameter, at a distance of 36 cm from the anal margin. It occupied 80 percent of the intestinal lumen. A biopsy collected during the examination suggested a diagnosis of neoplasia of vascular origin. After surgical resection, histopathological examination of the resected specimen confirmed the diagnosis of epithelioid hemangioma of the colon, which was backed up by the immunohistochemical panel (factor VIII, Ki-67, CD-34). At present, three years after the surgery, the patient is asymptomatic, she has recovered her normal weight and she has normal findings from control colonoscopy. Despite the rarity of neoplasia of vascular origin, this possibility should be considered in the differential diagnosis for colorectal tumors.

CONTEXTO: Hemangioma epitelióide ou hiperplasia angiolinfóide com eosinofilia são neoplasias vasculares benignas raras, habitualmente localizadas na face e pescoço. O acometimento do intestino grosso é excepcionalmente descrito, existindo apenas dois casos descritos na literatura mundial. O objetivo deste artigo é apresentar um caso de hemangioma epitelióide primário do sigmóide com diagnóstico histopatológico confirmado por meio de estudo imunoistoquímico. RELATO DE CASO: Mulher de 37 anos apresentou queixa de dor abdominal de forte intensidade, intermitente, localizada no hipogástrio. Dois meses após o início do quadro, notou alteração do hábito intestinal, evacuações com sangue, muco e perda ponderal de 4 quilos no período. Ao exame físico abdominal, identificou-se massa palpável no quadrante inferior esquerdo. Com suspeita clínica de neoplasia de cólon foi submetida a colonoscopia, que demonstrou presença de lesão vegetante de aproximadamente cinco centímetros de diâmetro, ocupando cerca de 80 por cento da luz colônica. A biópsia mostrou a presença de neoplasia de origem vascular. Após a ressecção cirúrgica, o exame histopatológico do espécime extirpado estabeleceu o diagnóstico de hemangioma epitelióide do cólon, confirmado por meio de painel imunoistoquímico (fator VIII, Ki-67, CD-34). No momento, a paciente encontra-se bem, tendo recuperado o peso inicial três anos após a cirurgia e apresenta resultado de colonoscopia de controle normal. Não obstante a raridade, deve-se considerar a possibilidade das neoplasias de origem vascular no diagnóstico diferencial dos tumores colorretais.