Intralesional corticosteroid treatment of periocular xanthogranuloma associated with adult-onset asthma

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Published Sep 27, 2023
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Vol. 101 No. 4 (2023): Tunis Med apr 2023

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Leila Knani
University of Sousse, Faculty of Medicine Ibn Jazzar Sousse, CHU Farhat Hached Sousse
Mehdi Oueslati
Université de Sousse, Faculté de médecine Ibn Jazzar Sousse, CHU Farhat Hached Sousse
https://orcid.org/0000-0003-1492-6827
Mohamed Ghachem
University of Sousse, Faculty of Medicine Ibn Jazzar Sousse, CHU Farhat Hached Sousse
Mohamed Ghorbel
University of Sousse, Faculty of Medicine Ibn Jazzar Sousse, CHU Farhat Hached Sousse

Abstract

Introduction: Adult-onset asthma and periocular xanthogranuloma (AAPOX) is a rare non-Langerhans cell histiocytic disorder.


Aim: To describe the periocular clinical findings in a patient diagnosed with AAPOX, treated successfully by intralesional corticosteroids.


Case Report: A 40-year-old woman presented with bilateral eyelid swelling and adult-onset asthma. Initial examination revealed bilateral yellow-orange, elevated, indurated, and nonulcerated masses at the upper eyelids. The laboratory data showed high level of IgG. Periocular biopsy samples showed xanthoma cells positive for CD68 and Touton giant cells. The patient received 2 intralesional 40 mg of triamcinolone acetonide with a local control. No complications were noted.


Commentaries: AAPOX is characterized by a histiocytic proliferation associated to an adult-onset asthma, systemic lymphadenopathy, salivary gland enlargement and elevated serum levels of IgG. Treatment options vary with no current consensus. Intralesional corticosteroids have been rarely reported in controlling the signs and symptoms of adult-onset xanthogranulomatous disease. Surgery was successful but demonstrated recurrence. Methotrexate has been proposed to treat refractory cases or as a potential corticosteroid-sparing therapy. In this case, intralesional corticosteroid was an effective and safe treatment for eyelid adult xanthogranuloma.

Keywords:

Adult onset asthma, Periocular xanthogranuloma, Orbit, Corticosteroid

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