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Canadian Respiratory Journal
Volume 16, Issue 5, Pages 159-162
Case Report

Clinical Manifestations and Treatment of Adult-Onset Asthma and Periocular Xanthogranuloma

Rodrigo Cavallazzi,1 Amyn Hirani,1 Tajender S Vasu,1 Robert C Sergott,2 Jurij R Bilyk,2 Ralph C Eagle,2 and Sandra Weibel1

1Division of Pulmonary and Critical Care Medicine, Thomas Jefferson University Hospital, Philadelphia, Pennsylvania, USA
2Wills Eye Institute, Department of Opthalmology, Thomas Jefferson University Hospital, Philadelphia, Pennsylvania, USA

Copyright © 2009 Hindawi Publishing Corporation. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.


BACKGROUND: Adult-onset asthma and periocular xanthogranuloma is an uncommon and recently described disease. Little is known about the condition because only a few case reports and series are available.

OBJECTIVE/METHODS: To describe the clinical manifestations, lung physiology, and response to systemic treatment of three patients with adult-onset asthma and periocular xanthogranuloma, followed by a review of the literature.

RESULTS: Three men, with an age at diagnosis ranging from 48 to 51 years, presented with right periorbital swelling, asthma and chronic rhinosinusitis. The patients’ lung physiology was consistent with airway obstruction. Diagnosis was established by periorbital biopsy. All patients received oral corticosteroids for their periorbital swelling, without significant clinical response. Two patients received oral methotrexate, with nearly complete resolution of periorbital swelling. A third patient received oral azathioprine, without clinical response. The three patients had improvement of their asthma with inhaled steroids/long-acting bronchodilator, and immunosuppressive medication.

CONCLUSION: A triad consisting of periorbital swelling, asthma and chronic rhinosinusitis should raise the suspicion of adult-onset asthma and periocular xanthogranuloma. Oral methotrexate should be considered as an alternative to corticosteroids in the treatment of this disorder.