Case Report: Non-episodic Angioedema With Eosinophilia in a Young Lactating Woman

Angioedema with eosinophilia is classified into two types: episodic angioedema with eosinophilia (EAE), known as Gleich's syndrome, and non-episodic angioedema with eosinophilia (NEAE). We present the case of a young lactating woman with non-episodic angioedema. She had no history of parasitic...

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Bibliographic Details
Published inFrontiers in immunology Vol. 12; p. 627360
Main Authors Hirmatsu-Ito, Mizuho, Nakamura, Nobuhisa, Miyabe, Megumi, Matsubara, Tatsuaki, Naruse, Keiko
Format Journal Article
LanguageEnglish
Published Switzerland Frontiers Media S.A 26.04.2021
Subjects
angioedema
case report
chemokine
cytokine
edema
eosinophil
Immunology
cytokine
angioedema
case report
chemokine
edema
eosinophil
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Summary:Angioedema with eosinophilia is classified into two types: episodic angioedema with eosinophilia (EAE), known as Gleich's syndrome, and non-episodic angioedema with eosinophilia (NEAE). We present the case of a young lactating woman with non-episodic angioedema. She had no history of parasitic or nonparasitic infections. Physical examination showed striking, non-pitting edema in both lower extremities. Her weight had not changed significantly throughout the course of the illness. She exhibited no other symptoms, and her vital signs were normal. There was no evidence of anemia, hypoalbuminemia, thyroid dysfunction, heart failure, renal failure, or postpartum cardiomyopathy. Based on these findings, we diagnosed her with angioedema with eosinophilia. Given the scarcity of information about this condition, we explored the dynamics between cytokines/chemokines and edema in this patient. We successfully quantified the edema by bioimpedance analysis. In addition, we revealed the involvement of interleukin-5 (IL-5), thymus- and activation-regulated chemokine/C-C motif chemokine ligand-17 (TARC/CCL-17), eotaxin-3/CCL-26, tumor necrosis factor-α (TNF-α), vascular endothelial growth factor (VEGF), monocyte chemotactic protein-4/CCL-13 (MCP-4/CCL-13), eotaxin-1/CCL-11, and regulated on activation, normal T expressed and secreted/CCL-5 (RANTES/CCL-5) in NEAE. Lastly, we elucidated the strong association between these parameters. To the best of our knowledge, this is the first such study of its kind.
Bibliography:Edited by: Lazaros Ignatios Sakkas, University of Thessaly, Greece
Reviewed by: Sabina Islam, Massachusetts General Hospital and Harvard Medical School, United States; Giuseppe Alvise Ramirez, Vita-Salute San Raffaele University, Italy
This article was submitted to Autoimmune and Autoinflammatory Disorders, a section of the journal Frontiers in Immunology
ISSN:1664-3224
1664-3224
DOI:10.3389/fimmu.2021.627360