Abstract Number: 216

A novel case of Morbihan disease successfully treated with Omalizumab

Miki Wada, Quynh Le, Matthew Howard, Adrian Mar

Meeting: 2023 Dermcoll

Session Information

Date: -

Session Title: Rosacea

Session Time: -

Aim: To present a case of treatment-refractory Morbihan disease successfully treated with Omalizumab.

Methods/Case Report: A 40-year-old female presented with a 2-year history of fixed facial erythema, induration and oedema affecting the chin, cheeks and eyelids, limiting vertical range of gaze. There were no papules or pustules and Pemberton’s sign was negative. She had no significant medical history, regular medications or allergies and no family history of connective tissue disorders. No allergic contact factors were identified. Clinical suspicion of Morbihan disease was confirmed with histopathology.

Her symptoms progressed despite 2 years of attempted treatment with numerous therapies including azaleic acid gel, brimonidine gel, oral minocycline, oral erythromycin, oral prednisolone and oral isotretinoin.

Given this lack of response, Omalizumab was commenced, with an initial dose of 450 mg and then 300 mg 4-weekly thereafter. Her symptoms mildly improved over 4 months. The dose was then increased to 450 mg 4-weekly, with significant reduction in facial erythema, induration and oedema, and secondarily marked improvement in vertical visual field range.

Results: The patient’s symptoms have improved and remain stable on Omalizumab 450 mg subcutaneous injections.

Conclusion: Morbihan disease can be notoriously difficult to treat. Not only is the persistent facial swelling and erythema physically disfiguring, but may also cause psychological distress.

Omalizumab is a recombinant monoclonal antibody against immunoglobulin E (IgE). It inhibits binding of IgE to mast cells and basophils, preventing histamine release. This raises the possibility that mast cells may play a role in Morbihan disease.

This is the second case of Morbihan disease treated successfully with Omalizumab. In 2019, Kafi et al report a similar case with complete resolution of facial swelling and erythema after 5 months of treatment with Omalizumab.

This report highlights Omalizumab as a potential treatment option for treatment-refractory Morbihan disease.