Acquired partial lipoatrophy as graft-versus-host disease and treatment with metreleptin: two case reports.
J Med Case Rep
; 12(1): 368, 2018 Dec 14.
Article
in En
| MEDLINE
| ID: mdl-30545408
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.Key words
Full text:
1
Collection:
01-internacional
Database:
MEDLINE
Main subject:
Leptin
/
Graft vs Host Disease
/
Lipodystrophy
Type of study:
Diagnostic_studies
Limits:
Adult
/
Female
/
Humans
Language:
En
Journal:
J Med Case Rep
Year:
2018
Document type:
Article
Affiliation country:
Japón