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Review
. 2024 Mar 1;52(2):23-31.
doi: 10.15586/aei.v52i2.978. eCollection 2024.

Omalizumab in the treatment of Morbihan syndrome in an adolescent girl - case report and literature review

Zofia Mazurek-Durlak  1 , Katarzyna Mularczyk  2 , Przemko Kwinta  2 , Grzegorz Lis  2 , Ewa Cichocka-Jarosz  2
Affiliations
Review

Omalizumab in the treatment of Morbihan syndrome in an adolescent girl - case report and literature review

Zofia Mazurek-Durlak et al. Allergol Immunopathol (Madr). .

Abstract

Morbihan syndrome (MS) is characterized by solid facial edema, usually related to rosacea or acne vulgaris. The facial edema deforms the patient's features, can impair peripheral vision, and affects quality of life. Its pathophysiology remains unclear. The disease usually has a slow and chronic course. MS most commonly affects middle-aged Caucasian men with rosacea and is rare in people below 20 years of age. MS is a diagnosis of exclusion. There is no standard treatment for MS, though systemic isotretinoin and antihistamines are mainly used. We present the case of an adolescent girl with MS nonresponding to 19 months of isotretinoin treatment with add-on antihistamines. Therapy with monthly administration of omalizumab (anti-IgE) for 6 months was an effective therapeutic option, improving the quality of life. Our case is the second description of omalizumab use in Morbihan syndrome, the first in an adolescent.

Keywords: Adolescent; Morbihan syndrome; anti-IgE; omalizumab; solid facial edema.

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References

    1. 1. Vasconcelos RC, Eid NT, Eid RT, Moriya FS, Braga BB, Michalany AO. Morbihan syndrome: A case report and literature review. An Bras Dermatol. 2016 Sep–Oct;91(5 Suppl 1):157–59. 10.1590/abd1806-4841.20164291 - DOI
    1. 2. Boparai RS, Levin AM, Lelli GJ Jr. Morbihan disease treatment: two case reports and a systematic literature review. Ophthalmic Plast Reconstr Surg. 2019 Mar/Apr;35(2):126–32. 10.1097/IOP.0000000000001229 - DOI
    1. 3. Mayur O, Martinez R, McNichol MC, McGee JS. Clinical and histological features and treatment outcomes of patients with Morbihan disease: A systematic review. Arch Dermatol Res. 2023 Apr 16;315:2427–429. 10.1007/s00403-023-02621-6 - DOI
    1. 4. Ramirez-Bellver JL, Pérez-González YC, Chen KR, Díaz-Recuero JL, Requena L, Carlson JA, et al. Clinicopathological and Immunohistochemical study of 14 cases of morbihan disease: an insight into its pathogenesis. Am J Dermatopathol. 2019 Oct;41(10):701–10. 10.1097/DAD.0000000000001378 - DOI
    1. 5. van der Linden MM, Arents BW, van Zuuren EJ. Diagnosis and treatment of Morbihan’s disease: a practical approach based on review of the Literature. J Clin Aesthet Dermatol. 2023 Oct;16(10):22–30.

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