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 1 , 2 , Hironori Shibata 2 , Takahiro Hosokawa 3 , Junichiro Iriehttp://orcid.org/https://orcid.org/0000-0003-2662-4121 4 , Hiroshi Ito 4 , and Tomonobu Hasegawa 2
  • 1 Department of Pediatrics, Saitama City Hospital 2460, Mimuro, Midori-ku Saitama-shi, Saitama 336-8522, Japan
  • 2 Division of Endocrinology and Metabolism, Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan
  • 3 Department of Radiology, Saitama Children’s Medical Center, Saitama, Japan
  • 4 Division of Endocrinology, Metabolism and Nephrology, Department of Internal Medicine, Keio University School of Medicine, Tokyo, Japan
Mayumi Hosokawa
  • Corresponding author
  • Department of Pediatrics, Saitama City Hospital 2460, Mimuro, Midori-ku Saitama-shi, Saitama 336-8522, Japan
  • Division of Endocrinology and Metabolism, Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan
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, Hironori Shibata
  • Division of Endocrinology and Metabolism, Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan
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, Takahiro Hosokawa, Junichiro IrieORCID iD: https://orcid.org/0000-0003-2662-4121, Hiroshi Ito
  • Division of Endocrinology, Metabolism and Nephrology, Department of Internal Medicine, Keio University School of Medicine, Tokyo, Japan
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and Tomonobu Hasegawa
  • Division of Endocrinology and Metabolism, Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan
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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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