Acquired partial lipoatrophy as graft-versus-host disease and treatment with metreleptin: Two case reports

Research output: Contribution to journalArticle

Abstract

Introduction: Acquired partial lipoatrophy has been reported after bone marrow transplantation during childhood; however, no adult cases have previously been reported. We herein report two adult cases of acquired partial lipoatrophy after transplantation. Case presentation: A 28-year-old Japanese woman developed diabetic ketoacidosis and received insulin therapy after bone marrow transplantation. She manifested partial lipoatrophy of the extremities, prominent insulin resistance, hyperglycemia, hypertriglyceridemia, and fatty liver. A 40-year-old Japanese woman underwent liver transplantation from a living donor for alcoholic liver disease after abstinence from alcohol. She newly developed non-alcoholic steatohepatitis and diabetes. Non-alcoholic steatohepatitis progressed to liver failure, and a second liver transplantation from a brain-dead donor was performed at 42 years of age. She demonstrated loss of subdermal fat of the upper and lower extremities, prominent insulin resistance, hyperglycemia, and hypertriglyceridemia. In both cases, the injection of recombinant methionyl human leptin reversed all of the metabolic abnormalities. Conclusions: Acquired partial lipoatrophy after transplantation is a manifestation of chronic graft-versus-host disease in adults. This entity is associated with diabetes with prominent insulin resistance and severe hypertriglycemia and can be successfully treated with metreleptin for the long term.

Original languageEnglish
Article number368
JournalJournal of Medical Case Reports
Volume12
Issue number1
DOIs
Publication statusPublished - Dec 14 2018

Fingerprint

Graft vs Host Disease
Fatty Liver
Insulin Resistance
Hypertriglyceridemia
Bone Marrow Transplantation
Hyperglycemia
Liver Transplantation
Transplantation
Alcohol Abstinence
Diabetic Ketoacidosis
Alcoholic Liver Diseases
Brain Death
Living Donors
Liver Failure
Lower Extremity
Therapeutics
Extremities
Fats
Tissue Donors
Insulin

Keywords

  • Diabetes
  • Insulin resistance
  • Leptin
  • Lipoatrophy
  • Transplantation

ASJC Scopus subject areas

  • Medicine(all)

Cite this

@article{15180cde6af544cc92d6929b08926cd3,
title = "Acquired partial lipoatrophy as graft-versus-host disease and treatment with metreleptin: Two case reports",
abstract = "Introduction: Acquired partial lipoatrophy has been reported after bone marrow transplantation during childhood; however, no adult cases have previously been reported. We herein report two adult cases of acquired partial lipoatrophy after transplantation. Case presentation: A 28-year-old Japanese woman developed diabetic ketoacidosis and received insulin therapy after bone marrow transplantation. She manifested partial lipoatrophy of the extremities, prominent insulin resistance, hyperglycemia, hypertriglyceridemia, and fatty liver. A 40-year-old Japanese woman underwent liver transplantation from a living donor for alcoholic liver disease after abstinence from alcohol. She newly developed non-alcoholic steatohepatitis and diabetes. Non-alcoholic steatohepatitis progressed to liver failure, and a second liver transplantation from a brain-dead donor was performed at 42 years of age. She demonstrated loss of subdermal fat of the upper and lower extremities, prominent insulin resistance, hyperglycemia, and hypertriglyceridemia. In both cases, the injection of recombinant methionyl human leptin reversed all of the metabolic abnormalities. Conclusions: Acquired partial lipoatrophy after transplantation is a manifestation of chronic graft-versus-host disease in adults. This entity is associated with diabetes with prominent insulin resistance and severe hypertriglycemia and can be successfully treated with metreleptin for the long term.",
keywords = "Diabetes, Insulin resistance, Leptin, Lipoatrophy, Transplantation",
author = "Yusuke Shibata and Atsuko Nakatsuka and Jun Eguchi and Satoshi Miyamoto and Yukari Masuda and Motoharu Awazawa and Akinobu Takaki and Ryuichi Yoshida and Takahito Yagi and Jun Wada",
year = "2018",
month = "12",
day = "14",
doi = "10.1186/s13256-018-1901-y",
language = "English",
volume = "12",
journal = "Journal of Medical Case Reports",
issn = "1752-1947",
publisher = "BioMed Central",
number = "1",

}

TY - JOUR

T1 - Acquired partial lipoatrophy as graft-versus-host disease and treatment with metreleptin

T2 - Two case reports

AU - Shibata, Yusuke

AU - Nakatsuka, Atsuko

AU - Eguchi, Jun

AU - Miyamoto, Satoshi

AU - Masuda, Yukari

AU - Awazawa, Motoharu

AU - Takaki, Akinobu

AU - Yoshida, Ryuichi

AU - Yagi, Takahito

AU - Wada, Jun

PY - 2018/12/14

Y1 - 2018/12/14

N2 - Introduction: Acquired partial lipoatrophy has been reported after bone marrow transplantation during childhood; however, no adult cases have previously been reported. We herein report two adult cases of acquired partial lipoatrophy after transplantation. Case presentation: A 28-year-old Japanese woman developed diabetic ketoacidosis and received insulin therapy after bone marrow transplantation. She manifested partial lipoatrophy of the extremities, prominent insulin resistance, hyperglycemia, hypertriglyceridemia, and fatty liver. A 40-year-old Japanese woman underwent liver transplantation from a living donor for alcoholic liver disease after abstinence from alcohol. She newly developed non-alcoholic steatohepatitis and diabetes. Non-alcoholic steatohepatitis progressed to liver failure, and a second liver transplantation from a brain-dead donor was performed at 42 years of age. She demonstrated loss of subdermal fat of the upper and lower extremities, prominent insulin resistance, hyperglycemia, and hypertriglyceridemia. In both cases, the injection of recombinant methionyl human leptin reversed all of the metabolic abnormalities. Conclusions: Acquired partial lipoatrophy after transplantation is a manifestation of chronic graft-versus-host disease in adults. This entity is associated with diabetes with prominent insulin resistance and severe hypertriglycemia and can be successfully treated with metreleptin for the long term.

AB - Introduction: Acquired partial lipoatrophy has been reported after bone marrow transplantation during childhood; however, no adult cases have previously been reported. We herein report two adult cases of acquired partial lipoatrophy after transplantation. Case presentation: A 28-year-old Japanese woman developed diabetic ketoacidosis and received insulin therapy after bone marrow transplantation. She manifested partial lipoatrophy of the extremities, prominent insulin resistance, hyperglycemia, hypertriglyceridemia, and fatty liver. A 40-year-old Japanese woman underwent liver transplantation from a living donor for alcoholic liver disease after abstinence from alcohol. She newly developed non-alcoholic steatohepatitis and diabetes. Non-alcoholic steatohepatitis progressed to liver failure, and a second liver transplantation from a brain-dead donor was performed at 42 years of age. She demonstrated loss of subdermal fat of the upper and lower extremities, prominent insulin resistance, hyperglycemia, and hypertriglyceridemia. In both cases, the injection of recombinant methionyl human leptin reversed all of the metabolic abnormalities. Conclusions: Acquired partial lipoatrophy after transplantation is a manifestation of chronic graft-versus-host disease in adults. This entity is associated with diabetes with prominent insulin resistance and severe hypertriglycemia and can be successfully treated with metreleptin for the long term.

KW - Diabetes

KW - Insulin resistance

KW - Leptin

KW - Lipoatrophy

KW - Transplantation

UR - http://www.scopus.com/inward/record.url?scp=85058603775&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85058603775&partnerID=8YFLogxK

U2 - 10.1186/s13256-018-1901-y

DO - 10.1186/s13256-018-1901-y

M3 - Article

C2 - 30545408

AN - SCOPUS:85058603775

VL - 12

JO - Journal of Medical Case Reports

JF - Journal of Medical Case Reports

SN - 1752-1947

IS - 1

M1 - 368

ER -