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)

Cutaneous tumors in pregnancy.

Pregnancy alters the frequency and natural course of certain skin tumors. Pregnancy-associated changes in melanocytic nevi are transient, and there is no substantiated evidence of increased risk of malignant transformation of melanocytic nevi in gestation. Characteristic vascular and pigment-related dermatoscopic features are helpful in evaluating pigmented lesions, but a biopsy should be performed for significant change or other worrisome features in a lesion. Outcomes for pregnancy-associated melanoma do not appear to be poorer compared with nonpregnancy melanoma; however, data are limited for advanced (stage III/IV) melanoma. Some studies suggest increased propensity for lymphovascular spread, but more data are needed for definitive conclusions and guidelines on prognostication, workup, and treatment of pregnancy-associated melanoma. Vascular tumors, particularly pyogenic granuloma (granuloma gravidarum), occur with increased frequency and are associated with pro-angiogenic hormonal influences. Dermatofibrosarcoma protuberans has a more aggressive course during pregnancy with both prompt surgical treatment and close monitoring for recurrence being indicated.

Increased expression of thymic stromal lymphopoietin and its receptor in Kimura's disease.

OBJECTIVE: The objective of this study was to investigate the expression of thymic stromal lymphopoietin (TSLP) and TSLP receptor (TSLPR) in Kimura's disease (KD). METHODS: Using parotid gland tissues from KD patients and control subjects, we quantified the expression levels of mRNA for TSLP, interleukin (IL)-25, IL-33, and their receptors by massively parallel sequencing. We also performed immunohistochemical analysis of TSLP and TSLPR, and counted cells immunoreactive for these proteins by the polymer immunocomplex and double immunofluorescence methods. RESULTS: The levels of mRNA for TSLP, TSLPR, and IL-25R, but not IL-25, IL-33, or IL-33R, were significantly elevated in parotid gland tissues from the KD group as compared to the control group. Immunohistochemical analysis revealed that TSLP- and TSLPR-positive cells were significantly increased in number in parotid gland tissues from KD patients. Double immunofluorescence staining showed that TSLP and TSLPR were localized mainly in CD68-positive macrophages and tryptase-positive mast cells, respectively. CONCLUSIONS: Overexpression of TSLP and TSLPR might contribute to the pathogenesis of KD through interactions between macrophages and mast cells. Regulation of TSLP/TSLPR signaling may be a potential therapeutic approach for KD.

Angiolymphoid hyperplasia with eosinophilia: a previously unreported complication of ear piercing.

Angiolymphoid hyperplasia with eosinophilia is a rare, benign vascular lesion characterized by discrete, painful papules. Although the exact etiology is unknown, trauma precedes many cases. We present a case of angiolymphoid hyperplasia with eosinophilia in the earlobes of a 15-year-old girl after ear piercing.

Angiolyphoid hyperplasia with eosinophilia occurring after venipuncture trauma.

Angiolymphoid hyperplasia with eosinophilia (ALHE), also known as epithelioid hemangioma, is a benign vascular proliferation of uncertain etiology. Herein, we present the first documented case of ALHE following venipuncture and review the published work regarding the relationship of this entity to preceding trauma.

Epitrochlear mass in a patient on maintenance hemodialysis--Kimura disease.

Kimura disease is a rare benign inflammatory disorder presenting as subcutaneous masses or lymphnodal mass in the cervical region. Kimura disease is reported sparsely in patients on maintenance hemodialysis. We report an unusual location of Kimura disease in a patient on maintenance hemodialysis, who had a prolonged, persistent asymptomatic eosinophilia.

Angiolymphoid hyperplasia with eosinophilia developing on an antecedent welding burn: a case report.

Angiolymphoid hyperplasia with eosinophilia (ALHE) describes a group of benign anomalous vascular hyperplasias which consist of epithelioid-like endothelial cells attached to dilated blood vessels, and infiltration of inflammatory cells, predominantly lymphocytes and some eosinophils. Here, we describe a healthy 34-year-old man, who had 10 well-defined, non-tender, red-to-brownish papules and subcutaneous nodules of 0.3-1.0 cm in diameter on his left forearm. The lesions started to appear about 4 months after an earlier electric welding rod burn had healed. The histopathologic diagnosis of the lesions was ALHE. Because the new lesions developed progressively and malignancy could not be excluded, the patient underwent a wide elliptical excision and received a split-thickness skin graft from his left thigh. His postoperative recovery was successful and has showed no evidence of recurrence after 5 years of follow-up. The forearm is an unusual site for ALHE; the antecedent burn was the key trigger for ALHE onset in this case.

Maladie de Kimura: a propos d'un cas

Introduction : La maladie de Kimura ou lymphogranulome eosinophile est une pathologie inflammatoire chronique tres rare; d'etiologie inconnue. Nous rapportons un cas de maladie de Kimura a localisation parotidienne et a travers une revue de la litterature; rappelons les principales caracteristiques cliniques; paracliniques; therapeutiques et evolutives de cette pathologie. Materiel et methodes : Patient de 17 ans qui a consulte devant l'apparition d'une tumefaction de la region parotidienne gauche evoluant depuis un an et sans paralysie faciale. Resultats : L'echographie cervico-parotidienne a revele une parotide gauche hypertrophiee siege de multiples nodules hypoechogenes. La tomodensitometrie cervico-faciale a mis en evidence une glande parotide gauche augmentee de taille et de structure heterogene nodulaire sans adenopathies cervicales. La cytopontion etait non concluante. Le patient a beneficie d'une parotidectomie exofaciale gauche et l'etude anatomopathologique de la piece operatoire revenue en faveur de la maladie de Kimura. Les suites operatoires etaient simples. Le recul est d'un an sans recidive. Discussion : La Maladie de Kimura se caracterise cliniquement par des nodules sous cutanes de localisation cervicofaciale; une augmentation du volume des glandes salivaires et des adenopathies satellites. Son diagnostic est histologique reposant sur l'identification d'une hyperplasie follicullaire avec des abces a polynucleaires eosinophiles. Le traitement est chirurgical le plus souvent mais dans certains cas la cortico- therapie est proposee. Ce diagnostic doit cependant rester a l'esprit devant toute masse cervico-faciale

Recurrent Kimura's disease: report of one case.

Kimura's disease (KD) is a rare, chronic inflammatory disease, and the etiology is not yet to be determined. The disease is usually found in young Asian men but rarely in Western population. Here, we report an 8-year-old boy with the initial presentation of right upper eyelid swelling. Laboratory tests disclosed peripheral eosinophilia and elevated IgE level. KD with lacrimal gland involvement was diagnosed. Surgical excision was performed several times and steroid was also used, however the disease recurred. Thereafter, steroid, cyclosporine-A and azathioprine were prescribed. The tumor mass resolved and the laboratory data returned to normal level.

Kimura disease.

Kimura disease is a benign rare chronic inflammatory disorder of unknown etiology that involves the lymph nodes and subcutaneous tissue of the head and neck regions. Elevated serum immunoglobulin E levels and peripheral blood eosinophilia are also common. This disease is most common in middle-aged Asian men. Although the etiology is unknown, it most probably represents an aberrant chronic immune response. Treatment for Kimura disease includes surgical resection and regional or systemic steroid therapy. Cytotoxic therapy and radiation have also been utilized. The disease has an excellent prognosis, although it may recur locally.

Hiperplasia angiolinfoide con eosinofilia: a propósito de dos casos / Angiolymphoid hyperplasia with eosinophilia: a propos of two cases

La Hiperplasia Angiolinfoide con Eosinofilia (HALE) es una proliferación vascular benigna, poco frecuente, que se caracteriza por nódulos localizados preferentemente en el cuero cabelludo, región periauricular y cuello, en adultos jóvenes. La histopatología muestra proliferación de vasos sanguíneos con endotelio prominente, acompañada de un infiltrado inflamatorio que incluye eosinófilos y linficitos. El tratamientos de elección es la resección quirúrgica; sin embargo, las recurrencias son frecuentes. Se presentan dos casos clínicos, se revisa la literatura y se discute la relación entre HALE y enfermedad de Kimura.

Kimura's disease involving thoracic and abdominal lymph nodes in a hemodialysis patient.

We report the first observed case of Kimura's disease occurring during hemodialysis, involving the thoracic and abdominal lymph nodes, which spontaneously resolved nearly completely after two months. Early diagnosis and recognition of Kimura's disease may spare both the patient and doctor from the need for unnecessary invasive diagnostic procedures. Therefore, we emphasize that we need to have a high index of suspicion regarding Kimura's disease.

Angiolymphoid hyperplasia with eosinophilia: evidence for a T-cell lymphoproliferative origin.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is commonly regarded an angioproliferative process characterized by the presence of prominent, bizarrely shaped blood vessels. These vessels are accompanied by an inflammatory infiltrate that is thought to be a reactive component. Both the cell of origin and the pathogenesis of ALHE remain controversial. To define the histogenesis of this disorder, we analyzed the phenotypic and genotypic profile of the inflammatory infiltrate in ALHE by immunohistochemistry and T-cell receptor gene rearrangement by polymerase chain reaction (PCR) and denaturing gradient gel electrophoresis, as well as automated high-resolution PCR fragment analysis. Five of 7 ALHE patients displayed a clonal T-cell population and proliferative T-cell activity in lesional tissue. Most of these cases followed a protracted and therapy-reluctant course with recurrences. These data suggest that ALHE or a subset of ALHE cases harboring a clonal T-cell population may represent a T-cell lymphoproliferative disorder of a benign or low-grade malignant nature.