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      Episodic angioedema associated with eosinophilia*

      case-report

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          Abstract

          We report a 12-year-old girl who presented with recurrent angioedema on the face, trunk, and extremities, and concomitant marked weight gain for 5 years. During the episode, her white blood cell count increased to 47.7×109/L with 89.9% eosinophils, followed by elevated serum level of IL-5, IgE, IgM, and LDH. Histopathology showed perivascular eosinophilic infiltration and diffuse eosinophilic infiltration throughout the dermis. Possible causes of hypereosinophilia and eosinophilic infiltration of vital organs were ruled out. We also tested the FIP1L1/PDGFRa and ETV6/PDGFRb fusion gene to exclude the possibility of myeloid and lymphatic vessel neoplasms. The patient was treated with methylprednisolone and discharged with an oral prednisolone taper, which resulted in complete remission of the edema and normalization of peripheral blood eosinophil count, serum IL-5 level, IgE, IgM, and LDH.

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          Episodic angioedema associated with eosinophilia.

          P. Kohler, I Sachs, Paul J. Marcoux … (1984)
          We studied four patients with recurrent attacks of angioedema, urticaria, and fever. During attacks, body weights increased up to 18 per cent, and leukocyte counts reached 108,000 per microliter (88 per cent eosinophils). The disease did not appear to threaten the function of vital organs. The two children received prednisone intermittently; the adults did not require treatment or were given alternate-day prednisone. Glucocorticoid therapy caused defervescence and diuresis and decreased total leukocyte and eosinophil counts. No patient had evidence of cardiac involvement (follow-up, 2 to 17 years). One patient remained in spontaneous remission for 20 years before symptoms recurred. Histologic studies showed that eosinophils localized and degranulated in the dermis, and they appeared to induce edema. Although this syndrome might be classified as a variant of the hypereosinophilic syndrome, we believe it is a separate entity because of its distinctive characteristics and its benign course.
          Article 0 comments Cited 36 times – based on 0 reviews     Review now
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            Nonepisodic Angioedema Associated with Eosinophilia: Report of 4 Cases and Review of 33 Young Female Patients Reported in Japan

            R Chikama, M Hosokawa, T Miyazawa … (1998)
            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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              Cytokine-associated angioedema syndromes including episodic angioedema with eosinophilia (Gleich's Syndrome).

              Javed Sheikh, Kevin Weller, Aleena Banerji (2006)
              Angioedema can be associated with many disorders and the presentation can be variable. Subsets of the angioedema syndromes are thought to be cytokine mediated (Table 1). Of these, the best described are the episodic angioedema with eosinophilia syndrome (Gleich's syndrome) and non-episodic angioedema with eosinophilia, which share some common features, but appear to have differences in pathophysiology. NERDS (nodules, eosinophilia, rheumatism, dermatitis and swelling), Clarkson syndrome (idiopathic capillary leak syndrome), and angioedema associated with aldesleukin (human recombinant IL-2) and IFN-alpha have also been reported in the literature, and have been discussed in this review. There is still much to be learned about the pathophysiology, diagnosis, and treatment of patients with these disorders. Our hope is that this review will be of help to those readers who care for patients with these disorders, and will stimulate interest in further research into the pathophysiology of these conditions.
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                Author and article information

                Journal
                Journal ID (nlm-ta): An Bras Dermatol
                Journal ID (iso-abbrev): An Bras Dermatol
                Journal ID (publisher-id): abd
                Title: Anais Brasileiros de Dermatologia
                Publisher: Sociedade Brasileira de Dermatologia
                ISSN (Print): 0365-0596
                ISSN (Electronic): 1806-4841
                Publication date (Print and electronic): Jul-Aug 2017
                Publication date (Print): Jul-Aug 2017
                Volume: 92
                Issue: 4
                Pages: 534-536
                Affiliations
                [1 ] Department of Dermatology, Jinling Hospital affiliated to Nanjing University School of Medicine - Jiangsu, China.
                Author notes
                Mailing address: Hong Sang, Department of Dermatology, Jinling Hospital affiliated to Nanjing University School of Medicine, Nanjing, 210002 Jiangsu, China. E-mail: sanghong@ 123456nju.edu.cn
                Article
                DOI: 10.1590/abd1806-4841.20174351
                PMC ID: 5595603
                SO-VID: 52ec5072-ebdd-4589-9045-251adadd449b
                License:

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial No Derivative License, which permits unrestricted non-commercial use, distribution, and reproduction in any medium provided the original work is properly cited and the work is not changed in any way.

                History
                Date received : 09 January 2015
                Date accepted : 09 April 2015
                Categories
                Subject: Case Report

                Keywords: interleukin-5,eosinophilia,angioedema
                Data availability:
                Keywords: interleukin-5, eosinophilia, angioedema

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