Evidence map›Paper›PMID 31314093›Full record

ArticleThe Journal of clinical endocrinology and metabolism2019

Comorbidities and Survival in Patients With Lipodystrophy: An International Chart Review Study.

Baris Akinci, Elif A Oral, Adam Neidert, Diana Rus, Wendy Y Cheng, Philippe Thompson-Leduc, Hoi Ching Cheung, Pamela Bradt, Maria Cristina Foss de Freitas, Renan Magalhães Montenegro and 3 more

Registry-linked trialOpen access · bronzeAbstract read
In one paragraph

Article in The Journal of clinical endocrinology and metabolism, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT02262806 (Compassionate Use of Metreleptin in Previously-Treated Patients With Partial Lipodystrophy), which is not on this map. Cited by 46 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
46citing papers in PubMed, 2 pooled it
2.4field-weighted citation impact, top 10% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT02262806 phase2active not recruitingnot on this map

Compassionate Use of Metreleptin in Previously-Treated Patients With Partial Lipodystrophy

TypeinterventionalSponsorNational Institute of Diabetes and Digestive and Kidney Diseases (NIDDK)Ran2014 to 2027Enrolled29ConditionsDiabetes, Lipodystrophy, HyperlipidemiaArmsMetreleptin
3 · Its place in the literature

Who cites it

46 citing papers in PubMed, 2 syntheses or guidelines pooled it, 81 citations in OpenAlex.

  1. Pooled it
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  4. Observational
  5. Review
  6. Article
  7. Insulin Receptor Family Autoantibodies in Patients with Type B Insulin Resistance.The Journal of clinical endocrinology and metabolism · 2026
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

13 authors at 7 institutions in 3 countries.

Baris AkinciDokuz Eylül University, Izmir, Turkey.
Elif A OralDivision of Metabolism, Endocrine & Diabetes and Brehm Center for Diabetes Research, Department of Internal Medicine, Michigan Medicine, University of Michigan, Ann Arbor, Michigan.
Adam NeidertDivision of Metabolism, Endocrine & Diabetes and Brehm Center for Diabetes Research, Department of Internal Medicine, Michigan Medicine, University of Michigan, Ann Arbor, Michigan.
Diana RusDivision of Metabolism, Endocrine & Diabetes and Brehm Center for Diabetes Research, Department of Internal Medicine, Michigan Medicine, University of Michigan, Ann Arbor, Michigan.
Wendy Y ChengAnalysis Group Inc., Boston, Massachusetts.
Philippe Thompson-LeducAnalysis Group Inc., Boston, Massachusetts.
Hoi Ching CheungAnalysis Group Inc., Boston, Massachusetts.
Pamela BradtAegerion Pharmaceuticals Inc., Cambridge, Massachusetts.
Maria Cristina Foss de FreitasUniversity of São Paulo-Ribeirao Preto Medical School, São Paulo, Brazil.
Renan Magalhães MontenegroFederal University of Ceará, Ceará, Brazil.
Virgínia Oliveira FernandesFederal University of Ceará, Ceará, Brazil.
Elaine CochranNational Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, Maryland.
Rebecca J BrownNational Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, Maryland.
Analysis Group (United States) · USMichigan Medicine · USNational Institutes of Health · USUniversidade Federal do Ceará · BRAegerion Pharmaceuticals (United States) · USDokuz Eylül University · TRUniversidade de São Paulo · BR

Funding

Molecular and Clinical Studies of Insulin ResistanceZIADK047050 · NIDDK · NATIONAL INSTITUTE OF DIABETES AND DIGESTIVE AND KIDNEY DISEASES · PI BROWN, REBECCA · 2009 to 2025
$6.1M
6 · The paper itself

Abstract

contextLimited natural history data are available in patients with non-HIV-related lipodystrophy syndromes who never received disease-specific therapies, making interpretation of benefits of therapies in lipodystrophy syndromes challenging.

objectiveWe assessed the natural history of non-HIV-related generalized lipodystrophy (GL) and partial lipodystrophy (PL) in patients who have never received leptin or other lipodystrophy-specific therapies. DESIGN/SETTING/PATIENTS: We conducted an international chart review of 230 patients with confirmed GL or PL at five treatment centers who never received leptin or other lipodystrophy-specific therapies. Patients were observed from birth to loss to follow-up, death, or date of chart abstraction. OUTCOME MEASURES: Lifetime prevalence of diabetes/insulin resistance and select organ abnormalities, time to diabetes/insulin resistance, first organ abnormality, disease progression, and mortality were described.

resultsDiabetes/insulin resistance was identified in 58.3% of patients. Liver abnormalities were the most common organ abnormality (71.7%), followed by kidney (40.4%), heart (30.4%), and pancreatitis (13.0%). Kaplan-Meier estimates of mean (SE) time to first organ abnormality were 7.7 years (0.9) in GL and 16.1 years (1.5) in PL (P < 0.001). Mean time to diabetes/insulin resistance was 12.7 years (1.2) in GL and 19.1 years (1.7) in PL (P = 0.131). Mean time to disease progression was 7.6 years (0.8) and comparable between GL and PL subgroups (P = 0.393). Mean time to death was 51.2 years (3.5) in GL and 66.6 years (1.0) in PL (P < 0.001).

conclusionsThis large-scale study provides comprehensive, long-term data across multiple countries on the natural history of non-HIV-related lipodystrophy.

Indexed as

AdolescentAdultAgedAge of OnsetComorbidityDiabetes ComplicationsDisease ProgressionFemaleGenetic TestingHumansInsulin ResistanceKaplan-Meier EstimateLipodystrophyLipodystrophy, Congenital GeneralizedMaleMiddle Aged

Identifiers

PMID31314093
PMCPMC6760298
OpenAlexW2958222855

What OpenQuestion holds

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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.