Acquired partial lipoatrophy as graft-versus-host disease and treatment with metreleptin: two case reports

Journal of Medical Case Reports

Ryuichi Yoshida¹  Takahito Yagi¹  Akinobu Takaki²  Yukari Masuda³  Motoharu Awazawa³  Jun Wada⁴  Jun Eguchi⁴  Satoshi Miyamoto⁴  Yusuke Shibata⁴  Atsuko Nakatsuka⁴ 

[1] Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences;Department of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences;Department of Metabolic Diseases, Graduate School of Medicine, The University of Tokyo;Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences;

关键词: Lipoatrophy;    Diabetes;    Transplantation;    Leptin;    Insulin resistance;   

DOI  :  10.1186/s13256-018-1901-y

来源: DOAJ

【 摘 要 】

Abstract Introduction Acquired partial lipoatrophy has been reported after bone marrow transplantation during childhood; however, no adult cases have previously been reported. We herein report two adult cases of acquired partial lipoatrophy after transplantation. Case presentation A 28-year-old Japanese woman developed diabetic ketoacidosis and received insulin therapy after bone marrow transplantation. She manifested partial lipoatrophy of the extremities, prominent insulin resistance, hyperglycemia, hypertriglyceridemia, and fatty liver. A 40-year-old Japanese woman underwent liver transplantation from a living donor for alcoholic liver disease after abstinence from alcohol. She newly developed non-alcoholic steatohepatitis and diabetes. Non-alcoholic steatohepatitis progressed to liver failure, and a second liver transplantation from a brain-dead donor was performed at 42 years of age. She demonstrated loss of subdermal fat of the upper and lower extremities, prominent insulin resistance, hyperglycemia, and hypertriglyceridemia. In both cases, the injection of recombinant methionyl human leptin reversed all of the metabolic abnormalities. Conclusions Acquired partial lipoatrophy after transplantation is a manifestation of chronic graft-versus-host disease in adults. This entity is associated with diabetes with prominent insulin resistance and severe hypertriglycemia and can be successfully treated with metreleptin for the long term.

【 授权许可】

Unknown