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

##plugins.themes.academic_pro.article.main##

Leila Knani
Mehdi Oueslati
Mohamed Ghachem
Mohamed Ghorbel

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

##plugins.themes.academic_pro.article.details##

References

  1. Jakobiec FA, Mills MD, Hidayat AA, et al. Periocular xanthogranulomas associated with severe adult-onset asthma. Trans Am Ophthalmol Soc 1993;91:99–125.
  2. Kerstetter J, Wang J. Adult Orbital Xanthogranulomatous Disease: A Review with Emphasis on Etiology, Systemic Associations, Diagnostic Tools, and Treatment. Dermatol Clin. 2015;33(3):457-63.
  3. London J, Soussan M, Gille T, Badelon I, Warzocha U, Galatoire O, Martin A, Dhote R, Abad S. Adult-onset asthma associated with periocular xanthogranuloma: new diagnostic and therapeutic approaches in a very rare systemic disease. Ophthalmic Plast Reconstr Surg. 2013;29(2):104-8.
  4. Maeng MM, Godfrey KJ, Jalaj S, Kazim M. Adult xanthogranulomatous disease of the orbit: case report of spontaneous regression and review of treatment modalities. Orbit. 2020;39(1):31-37.
  5. Papa BM, Prince HM, McNab AA, McKelvie P. IgG4-related ophthalmic disease in association with adult-onset asthma and periocular xanthogranuloma: a case report. Orbit. 2021; 7:1-6.
  6. Burris CK, Rodriguez ME, Raven ML, Burkat CN, Albert DM. Adult-Onset Asthma and Periocular Xanthogranulomas Associated with Systemic IgG4-Related Disease. Am J Ophthalmol Case Rep. 2016 Apr;1:34-37.
  7. Cavallazzi R, Hirani A, Vasu TS, Sergott RC, Bilyk JR, Eagle RC, Weibel S. Clinical manifestations and treatment of adult-onset asthma and periocular xanthogranuloma. Can Respir J. 2009;16(5):159-62.
  8. Elner VM, Mintz R, Demirci H, Hassan AS. Local corticosteroid treatment of eyelid and orbital xanthogranuloma. Ophthalmic Plast Reconstr Surg. 2006;22(1):36-40.
  9. Green MB, Daly MK, Laver NMV, Lefebvre DR. Adult-onset asthma and periocular xanthogranuloma - A rare infiltrative disease of the orbit and eyelid. Am J Ophthalmol Case Rep. 2021;22:101043.
  10. Asproudis I, Kanari M, Ntountas I, Ragos V, Goussia A, Batistatou A, Voulgari PV. Successful treatment with rituximab of IgG4-related disease coexisting with adult-onset asthma and periocular xanthogranuloma. Rheumatol Int. 2020;40(4):671-677.
  11. Singh M, Gautam N, Kaur M, Yadav D, Gupta P. Intravenous rituximab for the treatment of relapsing adult-onset asthma with periocular xanthogranuloma. Can J Ophthalmol. 2019;54(3):e115-e118.