Literature DB >> 31075084

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 Hosokawa1,2, Hironori Shibata2, Takahiro Hosokawa3, Junichiro Irie4, Hiroshi Ito4, Tomonobu Hasegawa2.   

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.

Entities:  

Keywords:  acquired lipodystrophy; graft-versus-host disease; hematopoietic stem cell transplantation; impaired glucose tolerance

Mesh:

Year:  2019        PMID: 31075084     DOI: 10.1515/jpem-2018-0356

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  4 in total

Review 1.  Body composition after allogeneic haematopoietic cell transplantation/total body irradiation in children and young people: a restricted systematic review.

Authors:  Ava Lorenc; Julian Hamilton-Shield; Rachel Perry; Michael Stevens
Journal:  J Cancer Surviv       Date:  2020-07-18       Impact factor: 4.442

Review 2.  Current Diagnosis, Treatment and Clinical Challenges in the Management of Lipodystrophy Syndromes in Children and Young People

Authors:  Samim Özen; Barış Akıncı; Elif A. Oral
Journal:  J Clin Res Pediatr Endocrinol       Date:  2019-08-22

Review 3.  Management of dyslipidemia after allogeneic hematopoietic stem cell transplantation.

Authors:  Yingxue Lu; Xiaojing Ma; Jie Pan; Rongqiang Ma; Yujie Jiang
Journal:  Lipids Health Dis       Date:  2022-08-02       Impact factor: 4.315

4.  Case Report: Metreleptin and SGLT2 Inhibitor Combination Therapy Is Effective for Acquired Incomplete Lipodystrophy.

Authors:  Ayako Nagayama; Kenji Ashida; Miki Watanabe; Kanoko Moritaka; Aya Sonezaki; Yoichiro Kitajima; Hirokazu Takahashi; Satoko Yoshinobu; Shimpei Iwata; Junichi Yasuda; Nao Hasuzawa; Shuichi Ozono; Seiichi Motomura; Masatoshi Nomura
Journal:  Front Endocrinol (Lausanne)       Date:  2021-05-31       Impact factor: 5.555

  4 in total

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