Jump to ContentJump to Main Navigation
Show Summary Details
More options …

Journal of Pediatric Endocrinology and Metabolism

Editor-in-Chief: Kiess, Wieland

Ed. by Bereket, Abdullah / Darendeliler, Feyza / Dattani, Mehul / Gustafsson, Jan / Luo, Fei Hong / Toppari, Jorma / Turan, Serap Demircioglu


IMPACT FACTOR 2018: 1.239

CiteScore 2018: 1.22

SCImago Journal Rank (SJR) 2018: 0.507
Source Normalized Impact per Paper (SNIP) 2018: 0.562

Online
ISSN
2191-0251
See all formats and pricing
More options …
Volume 32, Issue 5

Issues

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
  • 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
  • Email
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ Hironori Shibata
  • Division of Endocrinology and Metabolism, Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ 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
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ Tomonobu Hasegawa
  • Division of Endocrinology and Metabolism, Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
Published Online: 2019-05-10 | DOI: https://doi.org/10.1515/jpem-2018-0356

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.

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

References

  • 1.

    Misra A, Peethambaram A, Garg A. Clinical features and metabolic and autoimmune derangements in acquired partial lipodystrophy: report of 35 cases and review of the literature. Medicine 2004;83:18–34.PubMedCrossrefGoogle Scholar

  • 2.

    Misra A, Garg A. Clinical features and metabolic derangements in acquired generalized lipodystrophy: case reports and review of the literature. Medicine 2003;82:129–46.PubMedCrossrefGoogle Scholar

  • 3.

    Ogden CL, Li Y, Freedman DS, Borrud LG, Flegal KM. Smoothed percentage body fat percentiles for U.S. children and adolescents, 1999–2004. Natl Health Stat Report 2011;43:1–7.Google Scholar

  • 4.

    Rooney DP, Ryan MF. Diabetes with partial lipodystrophy following sclerodermatous chronic graft vs. host disease. Diabet Med 2006;23:436–40.CrossrefPubMedGoogle Scholar

  • 5.

    Adachi M, Asakura Y, Muroya K, Goto H, Kigasawa H. Abnormal adipose tissue distribution with unfavorable metabolic profile in five children following hematopoietic stem cell transplantation: a new etiology for acquired partial lipodystrophy. Clin Pediatr Endocrinol 2013;22:53–64.CrossrefPubMedGoogle Scholar

  • 6.

    Levy J, Burnett ME, Magro CM. Lipophagic panniculitis of childhood: a case report and comprehensive review of the literature. Am J Dermatopathol 2017;39:217–24.Web of SciencePubMedCrossrefGoogle Scholar

  • 7.

    Meacham LR, Sklar CA, Li S, Liu Q, Gimpel N, et al. Diabetes mellitus in long-term survivors of childhood cancer. Increased risk associated with radiation therapy: a report for the childhood cancer survivor study. Arch Intern Med 2009;169:1381–8.CrossrefWeb of SciencePubMedGoogle Scholar

  • 8.

    Kimura L, Alvarez G, Li N, Pawlikowska-Haddal A, Moore TB, et al. Temporary resolution of insulin requirement in acquired partial lipodystrophy associated with chronic graft-versus-host disease. Pediatr Blood Cancer 2017;64.Web of SciencePubMedGoogle Scholar

  • 9.

    Ceccarini G, Ferrari F, Santini F. Acquired partial lipodystrophy after bone marrow transplant during childhood: a novel syndrome to be added to the disease classification list. J Endocrinol Invest 2017;40:1273–4.PubMedCrossrefWeb of ScienceGoogle Scholar

  • 10.

    Diker-Cohen T, Cochran E, Gorden P, Brown RJ. Partial and generalized lipodystrophy: comparison of baseline characteristics and response to metreleptin. J Clin Endocrinol Metab 2015;100:1802–10.CrossrefWeb of SciencePubMedGoogle Scholar

  • 11.

    Brown RJ, Oral EA, Cochran E, Araujo-Vilar D, Savage DB, et al. Long-term effectiveness and safety of metreleptin in the treatment of patients with generalized lipodystrophy. Endocrine 2018;60:479–89.PubMedWeb of ScienceCrossrefGoogle Scholar

  • 12.

    Poglio S, Galvani S, Bour S, Andre M, Prunet-Marcassus B, et al. Adipose tissue sensitivity to radiation exposure. Am J Pathol 2009;174:44–53.CrossrefWeb of SciencePubMedGoogle Scholar

  • 13.

    Corvillo F, Aparicio V, Lopez-Lera A, Garrido S, Araujo-Vilar D, et al. Autoantibodies against perilipin 1 as a cause of acquired generalized lipodystrophy. Front Immunol 2018;9:2142.CrossrefPubMedWeb of ScienceGoogle Scholar

  • 14.

    Gokcebay DG, Azik F, Bayram C, Erdem AY, Fettah A, et al. Evaluation of endocrine and metabolic dysfunctions after hematopoietic stem cell transplantation in children: a study from Turkey. J Pediatr Endocrinol Metab 2017;30:683–91.PubMedWeb of ScienceCrossrefGoogle Scholar

  • 15.

    Kim YS, Kim MS, Kim SI, Lim SK, Lee HY, et al. Post-transplantation diabetes is better controlled after conversion from prednisone to deflazacort: a prospective trial in renal transplants. Transpl Int 1997;10:197–201.CrossrefPubMedGoogle Scholar

  • 16.

    Yamauchi T, Kamon J, Waki H, Terauchi Y, Kubota N, et al. The fat-derived hormone adiponectin reverses insulin resistance associated with both lipoatrophy and obesity. Nat Med 2001;7:941–6.PubMedCrossrefGoogle Scholar

About the article

Corresponding author: Mayumi Hosokawa, MD, Department of Pediatrics, Saitama City Hospital 2460, Mimuro, Midori-ku Saitama-shi, Saitama 336-8522, Japan; and Division of Endocrinology and Metabolism, Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan, Phone: +81-48-873-4111, Fax: +81-48-873-4111


Received: 2018-09-02

Accepted: 2019-02-18

Published Online: 2019-05-10

Published in Print: 2019-05-27


Author contributions: Mayumi Hosokawa, Hironori Shibata, Takahiro Hosokawa, Junichiro Irie, Hiroshi Ito and Tomonobu Hasegawa declare that they have no financial or personal relationships that could lead to a conflict of interest.

Research funding: None declared.

Employment or leadership: None declared.

Honorarium: None declared.

Competing interests: None declared

Research involving human subjects: This research was performed in accordance with the tenets of the Declaration of Helsinki.


Citation Information: Journal of Pediatric Endocrinology and Metabolism, Volume 32, Issue 5, Pages 537–541, ISSN (Online) 2191-0251, ISSN (Print) 0334-018X, DOI: https://doi.org/10.1515/jpem-2018-0356.

Export Citation

©2019 Walter de Gruyter GmbH, Berlin/Boston.Get Permission

Comments (0)

Please log in or register to comment.
Log in