Episodic angioedema with eosinophilia

Wolf, C.; Pehamberger, Hubert; Breyer, S; Leiferman, Kristin M.; Wolff, Klaus

doi:10.1016/s0190-9622(89)70003-7

Cited by 38 publications

(15 citation statements)

References 18 publications

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“…2 The histology of EAE usually reveals diffuse dermatitis with marked eosinophilic infiltration and dermal edema. 1,3 To the best of our knowledge, no previous cases of EAE in association with granulomatous lesions have been reported.…”

Section: Introductionmentioning

confidence: 96%

Histological Classification and Differential Diagnosis of Nonepisodic Angioedema With Eosinophilia

Goto

Ishizaki

Tanese

et al. 2016

The American Journal of Dermatopathology

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Nonepisodic angioedema with eosinophilia (NEAE) is a rare condition characterized with monoepisodic angioedema, a nonfebrile state, eosinophilia, normal serum IgM levels, and lack of internal organ involvement. The histology of this disease is not yet well known. The purpose of this study was to characterize the histopathologic features of NEAE. Twelve cases of clinically confirmed NEAE were retrieved from 6 institutions, and these cases were reviewed regarding the clinical data and histopathology, particularly regarding granulomatous lesions. The authors demonstrated that the histology of NEAE can be classified into 3 patterns that of eosinophilic granulomatous panniculitis (7/12 cases), eosinophilic dermatitis without granuloma formation (3/12 cases), and invisible dermatosis (2/12 cases). Six of the 7 granulomatous cases showed the characteristic eosinophilic granulomatous lesions containing individual necrotic adipocytes with membranous fat changes, which could be a differential clue to the diagnosis of NEAE. Review of the previously reported cases (n = 37) revealed that the histological classification could be adaptable to these reported cases. The authors should recognize the histological variation of NEAE and distinguish it from the histological mimickers, including eosinophilic granulomatosis with polyangiitis, erythema nodosum, hypereosinophilic syndrome, and episodic angioedema with eosinophilia.

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Section: Introductionmentioning

confidence: 96%

Histological Classification and Differential Diagnosis of Nonepisodic Angioedema With Eosinophilia

Goto

Ishizaki

Tanese

et al. 2016

The American Journal of Dermatopathology

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“…In addition, both epi sodic angioedema [9] and eosinophilic cellulitis of Wells and Smith [10] do not include the prominent nodules and rheumatic involvement which affected our patient.…”

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confidence: 72%

NERDS Syndrome: An Additional Case Report

et al. 1995

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NERDS is an eosinophilic disorder recently described by Butterfield and characterized by an association of nodules, eosinophilia, rheumatism, dermatitis and swelling. We describe an additional case, the third, of this new eosinophilic syndrome. The cardinal features included joint and cutaneous manifestations with prominent para-articular nodules and rheumatism, xerosis, recurrent urticarial eruption with angioedema associated with tissue and peripheral blood eosinophilia. A drug-induced (diclofenac) allergic rash and lymphadenopathy appeared during the course of the illness. Persistent leukocytosis with a maximum of 65% of eosinophils, mostly exhibiting the hypodense phenotype (activation index), was always present. During the acute phase of the disease, flow-cytometric analysis of blood and bone marrow revealed proliferation of activated CD4+/OKDR+ T helper cells and CD25+/OKDR+ eosinophils.

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“…Skin histology in 23 cases of non-EAE including 2 of our cases consisted of essentially the same features as those of EAE [1, 2, 3]with a diffuse or perivascular infiltration of eosinophils and lymphocytes of various degrees throughout the dermis. In 9 cases, the eosinophilic infiltration was also observed in the subcutaneous tissue, 4 of which showed a granulomatous tissue reaction consisting of eosinophils, lymphocytes, histiocytes and giant cells.…”

Section: Discussionmentioning

confidence: 98%

“…Since its original description by Gleich et al in 1984 [1], the number of cases of EAE has accumulated, confirming that this represents a true clinical entity [2, 3, 4, 5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26]. In a review of the Japanese literature, we found as many as 33 cases reported under the term of EAE [10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25].…”

Section: Discussionmentioning

confidence: 99%

“…Immunological studies suggest that activated T-cell-derived cytokines, mainly IL-5, were involved in the migration and activation of eosinophils in the skin [1, 2, 3, 10, 11, 12]. Indeed, some authors reported that among various cytokines such as IL-2, IL-3, IL-5, Eo-CFC, GM-CSF, only serum IL-5 was related to the disease activity and that it fluctuated in parallel with the peripheral blood eosinophilia and clinical symptoms [11].…”

Section: Discussionmentioning

confidence: 99%

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Nonepisodic Angioedema Associated with Eosinophilia: Report of 4 Cases and Review of 33 Young Female Patients Reported in Japan

et al. 1998

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Background: In 1984, Gleich et al. described 4 patients with episodic angioedema associated with eosinophilia (EAE), which was characterized by recurrent episodes of angioedema and urticaria, eosinophilia, elevated serum IgM, fever, increased body weight and a benign course without involvement of the internal organs demonstrating that it was a clinical entity distinct from the hypereosinophilic syndrome. Thereafter, 37 cases of EAE have been reported in Japan, 33 cases of which, although similar, had a different evolution from classical EAE. Objective: To describe 4 cases and review the cases of angioedema associated with eosinophilia reported in Japan. Results: Four Japanese female patients had persistent angioedema mainly involving the hands and lower legs, and eosinophilia which resolved within a few months. The review of the 37 cases of EAE in the Japanese literature demonstrated that in 33 cases, there were common characteristics which differed from EAE. These included: (1) the absence of recurrent attacks; (2) the predominance of young females (20–37 years, with a mean of 26 years); (3) the localization of the angioedema to the extremities; (4) the absence of increase in the serum IgM level, and (5) the effectiveness of low-dose prednisone or even the occurrence of spontaneous remission. Conclusion: We propose that persistent angioedema with eosinophilia can be classified into 2 types, i.e. one being an episodic (recurrent) type as reported by Gleich and a nonepisodic type as our 4 cases and others found in the Japanese literature.

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Episodic angioedema with eosinophilia

Cited by 38 publications

References 18 publications

Histological Classification and Differential Diagnosis of Nonepisodic Angioedema With Eosinophilia

Histological Classification and Differential Diagnosis of Nonepisodic Angioedema With Eosinophilia

NERDS Syndrome: An Additional Case Report

Nonepisodic Angioedema Associated with Eosinophilia: Report of 4 Cases and Review of 33 Young Female Patients Reported in Japan

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