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Oral ulcerations in a patient with autosomal dominant hyper-IgE syndrome (AD-HIES)

Authors :
Lawrence Ma
Johanna Borst
Source :
BMJ Case Rep
Publication Year :
2020
Publisher :
BMJ, 2020.

Abstract

A 23-year-old woman with autosomal dominant hyper-IgE syndrome complicated by recurrent pneumonia and sinusitis presented with 1 week of multiple painful oral ulcers unresponsive to empiric antiviral and antifungal treatment. Her ulcers progressively worsened and she required hospitalisation for intravenous hydration and pain control. PCR swab of an ulcer was positive for varicella-zoster virus. Her symptoms never fully resolved despite antiviral therapy, and within 2 weeks, she relapsed with new and worsening ulcers. Biopsy revealed chronic active inflammation with no evidence of viral inclusion bodies or fungal hyphae. She was diagnosed with recurrent aphthous stomatitis and referred to a local dentist for CO2 laser treatments with rapid resolution of her symptoms. This case highlights the broad differential for recurrent oral ulcers in people with a primary immunodeficiency. It also raises awareness of the benefits of laser therapy for aphthous stomatitis treatment and the importance of partnering with our colleagues in dentistry.

Details

ISSN :
1757790X
Volume :
13
Database :
OpenAIRE
Journal :
BMJ Case Reports
Accession number :
edsair.doi.dedup.....3719865f1da08d6870e42c3045af0573
Full Text :
https://doi.org/10.1136/bcr-2020-236705