Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with Eosinophilia

Nonepisodic angioedema with eosinophilia (NEAE) is a rare allergic disease with a young Japanese and East Asian female predominance. NEAE features transient, nonrecurrent angioedema and peripheral blood eosinophilia without visceral organ involvement. Angioedema in NEAE occurs on the extremities, wh...

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Main Authors: Takashi Hashimoto, Kanako Muneta, Ken Watanabe
Format: Article
Language:English
Published: Karger Publishers 2017-09-01
Series:Case Reports in Dermatology
Subjects:
Online Access:http://www.karger.com/Article/FullText/480398
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author Takashi Hashimoto
Kanako Muneta
Ken Watanabe
author_facet Takashi Hashimoto
Kanako Muneta
Ken Watanabe
author_sort Takashi Hashimoto
collection DOAJ
description Nonepisodic angioedema with eosinophilia (NEAE) is a rare allergic disease with a young Japanese and East Asian female predominance. NEAE features transient, nonrecurrent angioedema and peripheral blood eosinophilia without visceral organ involvement. Angioedema in NEAE occurs on the extremities, while the trunk and face are rarely involved. Here, we report a case of NEAE affecting only the face in an 80-year-old Japanese man. He was otherwise healthy and took no medication until the sudden development of angioedema on the face. The extremities and trunk were not involved. Skin biopsy examination revealed eosinophilic infiltration and degranulation between collagen bundles through the entire dermis, and perivascular and perifollicular infiltration of eosinophils and lymphocytes, but no evidence of vasculitis. Peripheral hypereosinophilia and high serum thymus- and activation-regulated chemokine (TARC) level were noted. Visceral organ involvement, parasite infection, and an allergic response were not detected in the patient. Administration of oral corticosteroid improved his symptoms rapidly and dramatically with improvements in the blood eosinophil count and serum TARC level. After the corticosteroid was discontinued, no recurrence was observed for 3 years. Thus, he was diagnosed as having NEAE. It should be noted that angioedema with eosinophilia might occur with an unusual presentation and might develop in elderly patients.
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spelling doaj.art-a0374f0a0faf4340be6a4edcfdcacea02022-12-22T01:46:01ZengKarger PublishersCase Reports in Dermatology1662-65672017-09-019316416810.1159/000480398480398Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with EosinophiliaTakashi HashimotoKanako MunetaKen WatanabeNonepisodic angioedema with eosinophilia (NEAE) is a rare allergic disease with a young Japanese and East Asian female predominance. NEAE features transient, nonrecurrent angioedema and peripheral blood eosinophilia without visceral organ involvement. Angioedema in NEAE occurs on the extremities, while the trunk and face are rarely involved. Here, we report a case of NEAE affecting only the face in an 80-year-old Japanese man. He was otherwise healthy and took no medication until the sudden development of angioedema on the face. The extremities and trunk were not involved. Skin biopsy examination revealed eosinophilic infiltration and degranulation between collagen bundles through the entire dermis, and perivascular and perifollicular infiltration of eosinophils and lymphocytes, but no evidence of vasculitis. Peripheral hypereosinophilia and high serum thymus- and activation-regulated chemokine (TARC) level were noted. Visceral organ involvement, parasite infection, and an allergic response were not detected in the patient. Administration of oral corticosteroid improved his symptoms rapidly and dramatically with improvements in the blood eosinophil count and serum TARC level. After the corticosteroid was discontinued, no recurrence was observed for 3 years. Thus, he was diagnosed as having NEAE. It should be noted that angioedema with eosinophilia might occur with an unusual presentation and might develop in elderly patients.http://www.karger.com/Article/FullText/480398EosinophiliaAngioedema with eosinophiliaNonepisodic angioedema with eosinophiliaThymus- and activation-regulated chemokine
spellingShingle Takashi Hashimoto
Kanako Muneta
Ken Watanabe
Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with Eosinophilia
Case Reports in Dermatology
Eosinophilia
Angioedema with eosinophilia
Nonepisodic angioedema with eosinophilia
Thymus- and activation-regulated chemokine
title Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with Eosinophilia
title_full Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with Eosinophilia
title_fullStr Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with Eosinophilia
title_full_unstemmed Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with Eosinophilia
title_short Facial Edema in an Elderly Man: An Unusual Presentation of Nonepisodic Angioedema with Eosinophilia
title_sort facial edema in an elderly man an unusual presentation of nonepisodic angioedema with eosinophilia
topic Eosinophilia
Angioedema with eosinophilia
Nonepisodic angioedema with eosinophilia
Thymus- and activation-regulated chemokine
url http://www.karger.com/Article/FullText/480398
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AT kenwatanabe facialedemainanelderlymananunusualpresentationofnonepisodicangioedemawitheosinophilia