Idiopathic lipoatrophic panniculitis in an adult treated with hydroxychloroquine and adipocyte grafting

• Published in: Annales de dermatologie et de vénéréologie Vol. 145; no. 11; p. 694
• Main Authors: Cornille, H, Adelmand, L, Garmi, R, Comoz, F, Verneuil, L
• Format: Journal Article
• Language: French
• Published: France 01.11.2018
• Subjects: Adipocytes - transplantation; Adult; Atrophy - therapy; Female; Humans; Hydroxychloroquine - therapeutic use; Lower Extremity; Panniculitis - therapy; Subcutaneous Fat - pathology; Panniculite lipoatrophiante; Autogreffe graisseuse; Hydroxychloroquine; Panniculite lipophagique; Annular lipoatrophy of the ankles; Lipoatrophic panniculitis; Lipoatrophie annulaire des chevilles; Fat grafting; Lipophagic panniculitis
• ISSN: 0151-9638
• DOI: 10.1016/j.annder.2018.07.013

Idiopathic lipoatrophic panniculitis is a rare disease. Various terms are used in the literature to describe similar entities: annular lipoatrophy of the ankles, lipophagic panniculitis and lipoatrophic panniculitis. In any event, it consists of an inflammatory process progressing to necrosis of the fatty tissue. Herein we report the case of a 37-year-old woman presenting characteristic signs: erythematous, annular, symmetrical, painful, inflammatory lesions of the lower extremities, progressing to areas of atrophy with an indurated inflammatory periphery. Histopathology showed lobular panniculitis composed primarily of lymphocytes and macrophages. The etiological investigations ruled out all other causes of lobular panniculitis. The patient was successfully treated with hydroxychloroquine 400mg/day for 1 year and she subsequently underwent fat grafting on the secondary lesions. Our literature review identified 23 reported cases of idiopathic lipoatrophic panniculitis since 2006, including 10 cases of annular ankle lipoatrophy. The most commonly used treatments are corticosteroids, hydroxychloroquine and methotrexate, with good efficacy being seen on the inflammatory phase. This disease seems a good indication for fat grafting to limit aesthetic sequelae. Lipoatrophic panniculitis, a rare disease, results in significant aesthetic sequelae which may be limited by early treatment initiation. The benefit-risk ratio of hydroxychloroquine suggests its value as first-line therapy in this pathology. Our case also demonstrates the value of filling of the secondary atrophy by fat grafting after the inflammatory period has ended.