Spontaneous Remission of Acquired Generalized Lipodystrophy Presenting in the Postpartum Period

Ranvir Bhatia, Prathyusha Chennupathi, Elliot D Rosenstein, Sonoo Advani
JCEM Case Reports, Volume 2, Issue 2, February 2024, luae009
https://doi.org/10.1210/jcemcr/luae009

Abstract

Acquired generalized lipodystrophy (AGL) is a rare condition characterized by the diffuse loss of adipose tissue resulting in hyperglycemia, severe insulin resistance, and sequelae of metabolic disease. Here we report the case of a 32-year-old woman who developed uncontrolled hyperglycemia and significant weight loss within 2 months postpartum. Upon endocrine evaluation, she was found to have generalized loss of adiposity, hypoleptinemia, and persistent hyperglycemia despite aggressive insulin administration. Glycemic response was obtained with U-500 intramuscular insulin, pioglitazone, and metformin-sitagliptin. At 14 months postpartum, the patient achieved spontaneous remission with normoglycemia off medication and restoration of adipose tissue deposition. Autoimmune workup revealed positive antinuclear antibodies (ANA) and anti–U1-ribonucleoprotein (anti–U1-RNP) titers, suggestive of an autoimmune etiology to her condition. This case of AGL represents the first reported case of spontaneous remission and the first to develop in the postpartum period.

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