Radicular cyst in primary dentition showing angiolymphoid hyperplasia with eosinophilia (ALHE)-like features: An immunohistochemical study

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)

Angiolymphoid Hyperplasia of the External Auditory Canal; Reconstruction of a Complex Defect.

Angiolymphoid hyperplasia (AH) was first described by Wells and Whimster in 1969 as a benign vasoproliferative pathology with a varied infiltrate of eosinophils, lymphocytes, and plasmatic cells. Clinical presentation has been described in the literature as small red-bluish nodules, less than 3 cm in diameter that can bleed in 25% of the cases and be pruritic and painful in 37% and 20% of the cases, respectively. Particularly, AH can appear in the ear; nevertheless, other regions have been affected, including the scalp, lips, tongue, orbit, muscle, and bone. Most of these cases have occurred in adults with an unknown etiology; however, an inflammatory process has been associated in approximately 20% with eosinophilia. No malignancy has been reported.

Angiolymphoid Hyperplasia with Eosinophilia in a Patient With Coccidioidomycosis.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare nodular mass that has not commonly been reported in the literature in association with coccidioidomycosis (CM). Coccidioidomycosis has other known skin manifestations including erythema nodosum and interstitial granulomatous dermatitis. Pulmonary CM is the most common form of the disease and the most common cause of CM-associated rash. This is an important clinical consideration for patients with ALHE who reside in CM-endemic areas, which notably include the southwestern region of the United States, Mexico, and South America. We report the case of an ALHE lesion that resolved following treatment for CM.

[Kimura's disease mimicking primary neoplasm of the parotid gland: A case report]. / Enfermedad de Kimura simulando neoplasia primaria de glándula parótida: Reporte de caso.

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.

Angiolymphoid hyperplasia: report of an unusual case in the oral cavity / Hiperplasia angiolinfoide: reporte de un caso inusual en cavidad oral

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.

Hiperplasia angiolinfoide con eosinofilia / Angiolymphoid Hyperplasia with eosinophilia

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)

Multiple Grouped Scalp Nodules in a Middle-Aged Man: A Rare Case of Angiolymphoid Hyperplasia with Eosinophilia.

An otherwise healthy man in his 50s presented complaining of pruritic lesions on the left side of his scalp. The lesions had slowly been growing in size over the preceding 30 years. They would occasionally bleed, and this is what ultimately prompted him to seek medical advice. Physical examination revealed multiple aggregated and soft, flesh-colored nodules on the left posterior auricular area of the scalp (Figure 1). No appreciable clinical lymphadenopathy was identified on examination. A shave biopsy of one of the nodules was performed for diagnostic clarification.

Angiolymphoid hyperplasia with eosinophilia versus Kimura's disease: a case report and a clinical and histopathological comparison.

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.

Angiolymphoid hyperplasia with eosinophilia versus Kimura's disease: a case report and a clinical and histopathological comparison

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.

Bilateral extraocular muscles enlargement from Kimura's disease of the orbit.

Kimura's disease (KD) is a rare chronic inflammatory disease of unclear etiology, characterized by subcutaneous nodules, mainly in the head and neck region, frequently associated with regional lymphadenopathy. Orbital involvement is infrequent and when it occurs, usually affects the eyelid or the lacrimal gland. We report a case of a 44-year-old man that presented with bilateral slowly progressive proptosis that was initially misdiagnosed as Graves' Ophthalmopathy. 15 months of worsening proptosis and the development of facial and temporal swelling led to further investigation. Computed tomography and magnetic resonance imaging showed enlargement of all recti muscles and diffuse orbital infiltration. An orbital biopsy was performed and was consistent with the diagnosis of KD. Long term oral corticosteroid showed marked improvement of proptosis and facial swelling. This case serves to emphasize that KD should be included in the differential diagnosis of inflammatory diseases of the orbit, even when characterized by predominant involvement of the extraocular muscles.

Glomus carotídeo bilateral. Reporte de un caso / Bilateral glomus caroticum. case report

Los glomus o paragangliomas carotídeos son tumores de crecimiento lento, hipervascularizados, poco frecuentes, derivados del cuerpo carotídeo. El objetivo es presentar un caso poco frecuente de tumores glómicos carotídeos bilaterales. Para su diagnóstico se realizó angiorresonancia magnética y angiografía de cuello. Se encontró una tumoración bilateral hipervascularizada a nivel de la división de la arteria carótida común, con signo de la lira...

Carotid glomus or paragangliomas are slow growing tumors, highly vascularized, rare, originated from the carotid body. The objective is to present a very rare case of bilateral carotid glomic tumors. Neck angioresonance was performed for diagnosis. A highly vascularized tumor was found bilaterally in the bifurcation of the common carotid artery, showing the lyre sign...

[An atypical form of Kimura's disease]. / Un cas de maladie de Kimura dans une forme atypique.

BACKGROUND: Kimura's disease is a rare benign chronic inflammatory disease of unknown aetiology. CASE REPORT: An 18-year-old atopic Brazilian patient consulted for recurrent facial eczema that improved slightly under topical corticosteroids. He presented a large infiltrated and eczematous subcutaneous plaque on the right supraorbital area, together with eyelid oedema. This was associated with complete right eyebrow alopecia, oedema of the upper lip and infiltrated facial papules. The tests showed hypereosinophilia (4000/mm(3)), a high IgE level (3786 kIU/L) and proteinuria (0.3g/24h). Histological examination revealed a lymphocytic eosinophil-rich inflammatory infiltrate in the superficial and deep dermis, with some lymphoid follicles in depth and proliferation of post-capillary venules. All of these elements led to the diagnosis of Kimura's disease. DISCUSSION: We report an unusual clinical form of Kimura's disease in a Brazilian patient. This disease has been classically described in young Asian men. In our case, a particularly large infiltrated and oedematous subcutaneous plaque was noted. In fact, the more common forms appear as subcutaneous nodules on the head and neck, which may be associated with locoregional adenopathy, involvement of the salivary glands, hypereosinophilia and raised serum IgE. In our case, the skin lesions, characterized by a large infiltrated and oedematous subcutaneous plaque, were unusual and could evoke IgG4-related disease. CONCLUSION: We report a case of Kimura's disease with an atypical presentation. The diagnosis was established by comparing certain histopathological features, and the possibility of IgG4-related disease was discussed.

Granuloma piógeno como diagnóstico diferencial de las masas del vestíbulo nasal / Pyogenic granuloma as differential diagnosis of nasal mass the hall: case report

Las masas del vestíbulo nasal son tumores poco frecuentes. Pueden dividirse en dos grupos; benignos como el papiloma invertido, angiofibroma juvenil y malignos como el carcinoma escamolacular. El granuloma piógeno corresponde a una hiperplasia inflamatoria con tejido de granulación de muy baja aparición. Se presenta el caso de una mujer de 34 años con masa nasal diagnosticada como carcinoma escamolacular del vestíbulo nasal, a quien se le realizo recesión amplia de la lesión...

The masses of the nasal vestibule are rare tumors. They can be divided into two groups: benign such as Inverted Papilloma and Juvenile Angiofibroma and malign as Escamolacular Carcinoma. Pyogenic granuloma corresponds to an inflammatory granulation tissue hyperplasia with very poor appearance. It is presented the case of a 34 year old woman diagnosed with nasal mass as Escamolacular Carcinoma of the nasal vestibule, a wide recession of the lesson was performed...

Hiperplasia angiolinfoide en espécimen de expansión tisular: Reporte de caso / Angiolymphoid hyperplasia in a tissue expansion specimen: A case report

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.