Bullous Wells' Syndrome: Case Report and Systematic Review
Abstract
Background/Objectives: Wells' syndrome (WS) is an uncommon cutaneous disease with unknown etiology. Itchy bullous lesions and erythematous plaques characterize the bullous WS (BWS), a rare subtype of the syndrome. We describe the case of a woman affected by chronic lymphocytic leukemia who developed BWS and responded to the classic corticosteroid treatment. We also systematically reviewed the literature, analyzing the clinical, laboratory, and histological features and treatments of this rare disease. Methods: We used the databases Ovid MEDLINE, PubMed, and EMBASE with the following search terms: ((bullous Wells' syndrome [MeSH Terms]) OR (eosinophilic cellulitis)) OR (bullous eosinophilic dermatitis) to identify and compare case reports of BWS. Results: We analyzed 28 patients, including our case. They were primarily female adults with a median age of 44.92 years. Blood eosinophilia was common, and histologically, the tissue samples showed an eosinophilic-neutrophilic dermal infiltrate. From a clinical perspective, the bullae were typically associated with or preceded by other lesions, primarily urticarial plaques, and mainly involved the extremities. Possible triggering agents of BWS include medications, insect bites, malignancies, and autoimmune/infectious diseases. Systemic steroids constituted the first-line treatment. Recent studies described the efficacy of the anti-interleukin-5 monoclonal antibody Mepolizumab in refractory cases of WS. Conclusions: Diagnosis of BWS…
How strong is the evidence?
Evidence Hub reads this as a systematic review, done in people. Its evidence level is 1 of 5 (Highest · pooled evidence). Pooled evidence from several studies sits at the top of the evidence pyramid. Reported size: 28 participants or records.
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