Acquired partial lipodystrophy with metabolic disease in children following hematopoietic stem cell transplantation

A report of two cases and a review of the literature

Mayumi Hosokawa, Hironori Shibata, Takahiro Hosokawa, Junichiro Irie, Hiroshi Ito, Tomonobu Hasegawa

Research output: Contribution to journalArticle

Abstract

Hematopoietic stem cell transplantation (HSCT) has been newly identified as an etiology underlying acquired lipodystrophy (ALD). We report about two children with leukemia who underwent HSCT and later manifested aberrant fat distributions consistent with acquired partial lipodystrophy (APL). Both patients manifested graft-versus-host disease (GVHD), suggesting that GVHD may trigger lipodystrophy. The patients exhibited diabetic blood glucose patterns in the oral glucose tolerance test (OGTT) with high homeostasis model assessment ratios (HOMA-Rs), hypertriglyceridemia, fatty liver, and decreased serum leptin and adiponectin levels. Both patients were diagnosed with APL with metabolic disease. A review of the data of patients with ALD after HSCT revealed common clinical features, including aberrant fat distribution, impaired glucose tolerance (IGT) or diabetes and dyslipidemia. Based on previous reports and our two cases, we speculate that GVHD in the adipose tissue supports the development of ALD after HSCT. In conclusion, children may develop APL after HSCT. Therefore, evaluations of fat distribution and metabolic disease may be important during the long-term follow-up of these patients.

Original languageEnglish
Pages (from-to)537-541
Number of pages5
JournalJournal of Pediatric Endocrinology and Metabolism
Volume32
Issue number5
DOIs
Publication statusPublished - 2019 May 1

Fingerprint

Hematopoietic Stem Cell Transplantation
Metabolic Diseases
Lipodystrophy
Graft vs Host Disease
Fats
Glucose Intolerance
Hypertriglyceridemia
Adiponectin
Fatty Liver
Glucose Tolerance Test
Dyslipidemias
Leptin
Blood Glucose
Adipose Tissue
Leukemia
Homeostasis
Lipodystrophy, Partial, Acquired
Serum

Keywords

  • acquired lipodystrophy
  • graft-versus-host disease
  • hematopoietic stem cell transplantation
  • impaired glucose tolerance

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Endocrinology, Diabetes and Metabolism
  • Endocrinology

Cite this

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abstract = "Hematopoietic stem cell transplantation (HSCT) has been newly identified as an etiology underlying acquired lipodystrophy (ALD). We report about two children with leukemia who underwent HSCT and later manifested aberrant fat distributions consistent with acquired partial lipodystrophy (APL). Both patients manifested graft-versus-host disease (GVHD), suggesting that GVHD may trigger lipodystrophy. The patients exhibited diabetic blood glucose patterns in the oral glucose tolerance test (OGTT) with high homeostasis model assessment ratios (HOMA-Rs), hypertriglyceridemia, fatty liver, and decreased serum leptin and adiponectin levels. Both patients were diagnosed with APL with metabolic disease. A review of the data of patients with ALD after HSCT revealed common clinical features, including aberrant fat distribution, impaired glucose tolerance (IGT) or diabetes and dyslipidemia. Based on previous reports and our two cases, we speculate that GVHD in the adipose tissue supports the development of ALD after HSCT. In conclusion, children may develop APL after HSCT. Therefore, evaluations of fat distribution and metabolic disease may be important during the long-term follow-up of these patients.",
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