Hypereosinophilia, defined as an absolute eosinophil count of >1,500/μL, can be caused by a number of allergic, infectious, paraneoplastic and neoplastic disorders. In cases of hypereosinophilia with lymphoid proliferation, pathological confirmation is essential to exclude either myeloid or lymphoid malignancy. A 38-year-old woman with both cervical lymphadenopathies and peripheral blood eosinophilia visited our clinic. She had already performed core biopsy of lymph nodes and diagnosed as Kimura disease at a regional hospital. At the time of our clinic visit, there were no palpable cervical lymph nodes. The blood test showed hypereosinophilia with a high total IgE level. There was no evidence of tissue infiltration of eosinophils except for duodenitis with eosinophilic infiltration. Based on these findings, she was diagnosed as Kimura disease. She treated with high-dose systemic corticosteroid (1 mg/kg) and additional immunosuppressants sequentially used cyclophosphamide and cyclosporine. However, her eosinophilia waxed and waned, and a left inguinal mass was newly found. Excisional biopsy findings showed large atypical lymphoid cells with numerous eosinophilis, and immunohistochemistry showed CD3+, CD20−, CD30+ and anaplastic lymphoma kinase (ALK). The final diagnosis was ALK-negative anaplastic large cell lymphoma. We report a case of anaplastic large cell lymphoma with marked peripheral eosinophilia misdiagnosed as Kimura disease. In the case of hypereosinophilia with lymphadenopathy, it is necessary to differentiate hematologic diseases through immunochemical staining.
Adult ; Ambulatory Care ; Angiolymphoid Hyperplasia with Eosinophilia* ; Biopsy ; Cyclophosphamide ; Cyclosporine ; Diagnosis ; Duodenitis ; Eosinophilia* ; Eosinophils ; Female ; Hematologic Diseases ; Hematologic Tests ; Humans ; Immunoglobulin E ; Immunohistochemistry ; Immunosuppressive Agents ; Lymph Nodes ; Lymphatic Diseases ; Lymphocytes ; Lymphoma ; Lymphoma, Large-Cell, Anaplastic* ; Phosphotransferases

Kimura disease is a rare idiopathic chronic inflammatory disorder. It typically presents in the head and neck area, whereas bilateral involvement is unusual. Its diagnosis requires it to be differentiated from other inflammatory diseases and from head and neck tumors. Treatment methods include conservative management, steroid administration, radiotherapy, and surgery; however, no single treatment of choice has been established. Herein, we report an unusual presentation of Kimura disease with bilateral parotid involvement. This case was treated by surgical excision.
Angiolymphoid Hyperplasia with Eosinophilia* ; Diagnosis ; Head ; Head and Neck Neoplasms ; Neck ; Parotid Neoplasms ; Radiotherapy

Kimura disease is a rare idiopathic chronic inflammatory disorder. It typically presents in the head and neck area, whereas bilateral involvement is unusual. Its diagnosis requires it to be differentiated from other inflammatory diseases and from head and neck tumors. Treatment methods include conservative management, steroid administration, radiotherapy, and surgery; however, no single treatment of choice has been established. Herein, we report an unusual presentation of Kimura disease with bilateral parotid involvement. This case was treated by surgical excision.
Angiolymphoid Hyperplasia with Eosinophilia* ; Diagnosis ; Head ; Head and Neck Neoplasms ; Neck ; Parotid Neoplasms ; Radiotherapy

Kimura's disease is a rare, benign, slow-growing chronic inflammatory swelling with a predilection for the head and neck region and is almost always with peripheral blood eosinophilia and elevated serum IgE levels. It is endemic in Asian males and rare in Western people. Surgical excision of the lesion is the first line therapy. Drug and radiation therapy have to be considered for the refractory lesions.
Angiolymphoid Hyperplasia with Eosinophilia ; diagnosis ; Asian Continental Ancestry Group ; Eosinophilia ; pathology ; Humans ; Immunoglobulin E ; blood ; Inflammation ; pathology ; Male ; Neck ; pathology

A 48-year-old middle aged male presented swelling lymph nodes and mass in neck for 5 years. Physical examination shows swollen mass in head and neck regions. The masses could be touched in bilateral parotids and neck with a little movement and moderate tenderness. The level of IgG was normal, but Eosinophi count was high. The function of heart liver and kidney was normal. The result of B-mode ultrasonography reveals bilateral parotids and subcutaneous near parotids were widely swollen and several swollen lymph nodes in neck. pathological examination displays features of a large number of lymph follicles hyperplasia, acidophilic granulocyte infiltration, capillary hyperplasia and fibrosis of different level. The disease were eventually diagnosed by pathological examination. Method of treatment includes glucocorticoid drug therapy, surgical resection and local radiotherapy. The last treatment of patients with Kimura's disease should be combined with the clinical manifestation of them to determine the individualized treatment, so as to improve the quality of life of patients.
Angiolymphoid Hyperplasia with Eosinophilia ; diagnosis ; pathology ; therapy ; Glucocorticoids ; therapeutic use ; Humans ; Lymph Nodes ; pathology ; Male ; Middle Aged ; Neck ; Parotid Gland ; pathology

OBJECTIVETo study the clinicopathologic features, diagnosis and differential diagnosis of epithelioid hemangioma.

METHODSThe morphologic features of 7 cases of epithelioid hemangioma of skin, bone and venous vessels were studied.

RESULTSThere were altogether 4 male and 3 female patients (median age = 34 years; age range from 14 to 54 years). The 3 skin cases presented as single or multiple erythematous to bluish nodules or papules, with or without itchiness. The 2 bone cases appeared as osteolytic expansile lesions on radiologic examination. The remaining 2 cases involved medium-sized venous structures and presented as small isolated nodules in soft tissue. Histologically, the lesions were characterized by the presence of exuberant endothelial proliferations with various degree of inflammatory reaction. The neoplastic endothelial cells were plump, eosinophilic and polygonal, forming vascular channels. Occasional solid sheet-like arrangement was demonstrated. Intracytoplasmic vacuoles were commonly identified, indicating formation of primary lumen. The surrounding stroma contained various number of eosinophils and lymphoplasmacytic cells. Immunohistochemical study showed that the tumor cells were positive for endothelial markers (CD31 and CD34) and negative for epithelial marker (cytokeratin). Follow-up information was available in 6 cases. The duration of follow-up ranged from 5 to 36 months (median = 14 months). There was no evidence of recurrence or distant metastasis.

CONCLUSIONSEpithelioid hemangioma is a rare benign curable lesion which can be multifocal, involving skin, soft tissue and bone. It needs to be distinguished from Kimura's disease and epithelioid hemangioendothelioma.

Adolescent ; Adult ; Angiolymphoid Hyperplasia with Eosinophilia ; pathology ; Antigens, CD34 ; metabolism ; Bone Neoplasms ; metabolism ; pathology ; surgery ; Diagnosis, Differential ; Female ; Follow-Up Studies ; Hemangioendothelioma, Epithelioid ; pathology ; Hemangioma ; metabolism ; pathology ; surgery ; Humans ; Male ; Middle Aged ; Platelet Endothelial Cell Adhesion Molecule-1 ; metabolism ; Skin Neoplasms ; metabolism ; pathology ; surgery

A 35-year-old woman presented with history of a painless, slow-growing nodule in a left eye caruncle over the last 2 months. The visual acuity was 20 / 20 and the ocular movements were in the normal range. The venereal disease research laboratory test, erythrocyte sedimentation rate, serum angiotensin converting enzyme level, and chest radiograph were all normal. An excisional biopsy was done to confirm the diagnosis. A 1.3 x 0.5 x 0.3 cm sized nodule was extracted and sent for histopathologic examination. Hematoxylin-eosin staining showed follicular hyperplasia with perifollicular fibrosis, an eosinophil infiltrate, and proliferation of capillary vessels. The capillaries were lined by normal-appearing, flat, spindle-shaped endothelial cells. On the basis of these histopathologic findings, the diagnosis of Kimura disease in a caruncle was established. This is the first report describing Kimura disease localized to a caruncle. Kimura disease should be suspected and included in the differential diagnosis of a caruncular mass lesion.
Adult ; Angiolymphoid Hyperplasia with Eosinophilia/*diagnosis ; Asian Continental Ancestry Group ; Conjunctiva/*pathology ; Conjunctival Diseases/*diagnosis ; Diagnosis, Differential ; Female ; Humans

A 35-year-old woman presented with history of a painless, slow-growing nodule in a left eye caruncle over the last 2 months. The visual acuity was 20 / 20 and the ocular movements were in the normal range. The venereal disease research laboratory test, erythrocyte sedimentation rate, serum angiotensin converting enzyme level, and chest radiograph were all normal. An excisional biopsy was done to confirm the diagnosis. A 1.3 x 0.5 x 0.3 cm sized nodule was extracted and sent for histopathologic examination. Hematoxylin-eosin staining showed follicular hyperplasia with perifollicular fibrosis, an eosinophil infiltrate, and proliferation of capillary vessels. The capillaries were lined by normal-appearing, flat, spindle-shaped endothelial cells. On the basis of these histopathologic findings, the diagnosis of Kimura disease in a caruncle was established. This is the first report describing Kimura disease localized to a caruncle. Kimura disease should be suspected and included in the differential diagnosis of a caruncular mass lesion.
Adult ; Angiolymphoid Hyperplasia with Eosinophilia/*diagnosis ; Asian Continental Ancestry Group ; Conjunctiva/*pathology ; Conjunctival Diseases/*diagnosis ; Diagnosis, Differential ; Female ; Humans

PURPOSE: The authors of the present study describe a rare case of angiolymphoid hyperplasia with eosinophilia (ALHE) of the eyelid. CASE SUMMARY: A 63-year-old male who was diagnosed with ALHE based on biopsy of an inguinal mass presented with an eyelid mass of 1 month duration. A light brown, solitary, 1.0 x 0.5 cm-sized mass involved the right upper eyelid. There was no lymphadenopathy, but eosinophilia was present. An excisional biopsy of the mass was performed for diagnosis and management. Macroscopic examination of the excised mass revealed a well-defined, smooth, firm, yellowish-red colored lesion measuring 1.0 x 0.6 x 0.5 cm. Histopathology showed the proliferation of small blood vessels, many of which were lined by enlarged endothelial cells with uniform ovoid nuclei and intracytoplasmic vacuoles. The distinctive endothelial cells were described as having a cobblestone appearance. In addition, a perivascular and interstitial infiltrate composed primarily of lymphocytes and eosinophils was present. ALHE was finally confirmed with clinical and microscopic examination. CONCLUSIONS: The authors of the present study report a rare case of ALHE of the eyelid and suggest that a differential diagnosis should be considered.
Angiolymphoid Hyperplasia with Eosinophilia ; Biopsy ; Blood Vessels ; Diagnosis, Differential ; Endothelial Cells ; Eosinophilia ; Eosinophils ; Eyelids ; Humans ; Light ; Lymphatic Diseases ; Lymphocytes ; Male ; Middle Aged ; Vacuoles

Objective: To describe a rare case of Kimura’s disease initially misdiagnosed as malignancy then tuberculosis. Methods: Design: Case report Setting: Tertiary Private Hospital Patient: One Results: A 30-year-old male with a 6-year history of gradually-enlarging right infra-auricular mass revealed an enlarged mass in the right infraauricular area and multiple cervical lymphadenopathies on physical examination. Initial fine-needle aspiration biopsy was interpreted as pleomorphic adenocarcinoma but succeeding work-ups and imaging studies led to treatment for tuberculosis. Subsequent biopsies finally led to the proper histopathologic diagnosis of Kimura’s disease and the patient was shifted to appropriate treatment with oral prednisone. Conclusion Kimura’s disease is rare and may be confused with other diseases such as malignancy or tuberculosis. Histopathologic diagnosis is necessary as its treatment differs from tuberculosis and other diseases.
Human ; Male ; Adult ; ANGIOLYMPHOID HYPERPLASIA WITH EOSINOPHILIA ; TUBERCULOSIS ; PHYSICAL EXAMINATION ; DIAGNOSIS ; DIAGNOSTIC TECHNIQUES AND PROCEDURES BIOPSY, FINE-NEEDLE ; BIOPSY ; PREDNISONE ; Diagnostic Errors ; history ; diagnosis