Morbihan Disease May Induce Nonspecific Inflammatory of Extra-Facial Region: A Case Report and Literature Review.

This case describes a 29-year-old man with Morbihan disease, characterized by a 5-year history of recurrent facial erythema, papules, and pustules, followed by persistent right face edema. Initially misdiagnosed as lupus erythematosus, he received multiple treatments with only transient relief. Clinical manifestations combined with pathological findings confirmed a diagnosis of Morbihan disease associated with rosacea. Notably, the patient developed erythema and papules on the inner thighs, along with small papules on the trunk and upper limbs for 3 months. A biopsy revealed nonspecific inflammation. Since the morphology of these lesions differed from any known skin disease and no other cause could be identified, we believe this may be a manifestation of the disease. Extra-facial manifestation not previously reported. Various therapies were attempted: doxycycline and azelaic acid improved erythema; baricitinib was ineffective; while omalizumab and intense pulsed light (IPL) achieved partial improvement in erythema and edema. Long-term isotretinoin therapy provided sustained remission without relapse during tapering, confirming its efficacy and safety. This case expands the clinical spectrum of Morbihan disease by documenting extra-facial involvement and reported isotretinoin as an effective long-term therapy, with omalizumab and IPL as promising adjuncts. Furthermore, the literature review (2004-2025) summarizes the clinical features, pathology, and treatment responses of reported cases, showing a male-to-female ratio of 2.81:1. Lymphatic or vascular dilation was significantly correlated with a favorable outcome. Conversely, dermal edema and granuloma were inversely associated with treatment efficacy, indicating poorer prognosis. However, based on uncontrolled data, these findings are strictly hypothesis-generating and do not establish a definitive treatment hierarchy.