Evidence map›Paper›PMID 31233501›Full record

ArticlePLoS genetics2019

Congenital lipodystrophy induces severe osteosclerosis.

Wei Zou, Nidhi Rohatgi, Jonathan R Brestoff, Yan Zhang, Erica L Scheller, Clarissa S Craft, Michael D Brodt, Nicole Migotsky, Matthew J Silva, Charles A Harris and 1 more

Open access · goldAbstract read
In one paragraph

Article in PLoS genetics, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers.

0numbers the graph read from it
0cells of the map it votes in
34citing papers in PubMed
2.2field-weighted citation impact, top 11% 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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

34 citing papers in PubMed, 43 citations in OpenAlex.

  1. Article
  2. Review
  3. Article
  4. Article
  5. Article
  6. Depletion of marrow adipo-CAR cells in mice enhances bone formation by activating bone morphogenetic protein receptor (BMPR) in pre-osteoblasts.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2025
    Article
  7. Article
  8. Bone marrow adipocytes: key players in vascular niches, aging, and disease.Frontiers in cell and developmental biology · 2025
    Review
  9. Review
  10. Lipolysis products from triglyceride-rich lipoproteins induce stress protein ATF3 in osteoblasts.Journal of orthopaedic research : official publication of the Orthopaedic Research Society · 2024
    Article
  11. Article
  12. The Multifaceted Roles of Bone Marrow Adipocytes in Bone and Hematopoietic Homeostasis.The Journal of clinical endocrinology and metabolism · 2023
    Review
  13. Observational
  14. Article
  15. Review
  16. Chondroprotection of leptin deficiency demystified?Osteoarthritis and cartilage · 2023
    Article
  17. Article
  18. Article
  19. Article
  20. Article
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

11 authors at 1 institution in 2 countries.

Wei ZouDivision of Anatomic and Molecular Pathology, Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, United States of America.
Nidhi RohatgiDivision of Anatomic and Molecular Pathology, Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, United States of America.
Jonathan R BrestoffDivision of Laboratory and Genomic Medicine, Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, United States of America.
Yan ZhangDivision of Anatomic and Molecular Pathology, Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, United States of America.
Erica L SchellerDivision of Bone and Mineral Diseases, Department of Medicine, Washington University School of Medicine, St. Louis, MO, United States of America.
Clarissa S CraftDivision of Bone and Mineral Diseases, Department of Medicine, Washington University School of Medicine, St. Louis, MO, United States of America.
Michael D BrodtDepartment of Orthopedic Surgery, Washington University School of Medicine, St. Louis, MO, United States of America.
Nicole MigotskyDepartment of Orthopedic Surgery, Washington University School of Medicine, St. Louis, MO, United States of America.ORCID 0000-0002-8478-4281
Matthew J SilvaDepartment of Orthopedic Surgery, Washington University School of Medicine, St. Louis, MO, United States of America.ORCID 0000-0001-7375-4522
Charles A HarrisDivision of Endocrinology, Metabolism, and Lipid Research, Department of Medicine, Washington University School of Medicine, St. Louis, MO, United States of America.ORCID 0000-0003-1815-5046
Steven L TeitelbaumDivision of Anatomic and Molecular Pathology, Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, United States of America.ORCID 0000-0002-4054-6679
Washington University in St. Louis · US

Funding

Washington University Nutrition Obesity Research CenterP30DK056341 · NIDDK · WASHINGTON UNIVERSITY · PI Dominic N Reeds · 1999 to 2026
$30.2M
WU P&FP30DK020579 · NIDDK · WASHINGTON UNIVERSITY · PI Clay F. Semenkovich · 2013 to 2026
$27.1M
Response of the Osteoporotic Skeleton to Mechanical LoadingR01AR047867 · NIAMS · WASHINGTON UNIVERSITY · PI MATTHEW J SILVA · 2001 to 2026
$8.4M
Resource Based Center for Musculoskeletal Biology and Medicine (Overall Application)P30AR074992 · NIAMS · WASHINGTON UNIVERSITY · PI MATTHEW J SILVA · 2019 to 2026
$6.8M
The Role of the Osteoblast Secretome in Bone FormationP30AR057235 · NIAMS · WASHINGTON UNIVERSITY · PI SILVA, MATTHEW J · 2009 to 2018
$5.7M
Mechanisms of AVB3 Integrin Mediated Bone ResorptionR37AR046523 · NIAMS · WASHINGTON UNIVERSITY · PI TEITELBAUM, STEVEN L · 2010 to 2019
$3.3M
FAT TALKS TO BONER01DK111389 · NIDDK · WASHINGTON UNIVERSITY · PI TEITELBAUM, STEVEN L · 2017 to 2021
$1.9M
GLUCOCORTICOID RECEPTOR POST-TRANSLATIONAL MODIFICATIONS IN INSULIN RESISTANCER01DK106083 · NIDDK · WASHINGTON UNIVERSITY · PI SEMENKOVICH, CLAY F. · 2016 to 2020
$1.7M
Neural regulation of skeletal biology and periodontal disease progression in typeR00DE024178 · NIDCR · WASHINGTON UNIVERSITY · PI SCHELLER, ERICA LYNN · 2016 to 2018
$735k
NIAMS NIH HHS P30 AR057235NIAMS NIH HHS P30 AR074992NIAMS NIH HHS R01 AR047867NIAMS NIH HHS R37 AR046523NIDCR NIH HHS R00 DE024178NIDDK NIH HHS P30 DK020579NIDDK NIH HHS P30 DK056341NIDDK NIH HHS R01 DK106083NIDDK NIH HHS R01 DK111389
6 · The paper itself

Abstract

Berardinelli-Seip congenital generalized lipodystrophy is associated with increased bone mass suggesting that fat tissue regulates the skeleton. Because there is little mechanistic information regarding this issue, we generated "fat-free" (FF) mice completely lacking visible visceral, subcutaneous and brown fat. Due to robust osteoblastic activity, trabecular and cortical bone volume is markedly enhanced in these animals. FF mice, like Berardinelli-Seip patients, are diabetic but normalization of glucose tolerance and significant reduction in circulating insulin fails to alter their skeletal phenotype. Importantly, the skeletal phenotype of FF mice is completely rescued by transplantation of adipocyte precursors or white or brown fat depots, indicating that adipocyte derived products regulate bone mass. Confirming such is the case, transplantation of fat derived from adiponectin and leptin double knockout mice, unlike that obtained from their WT counterparts, fails to normalize FF bone. These observations suggest a paucity of leptin and adiponectin may contribute to the increased bone mass of Berardinelli-Seip patients.

Indexed as

AdipocytesAdiponectinAdipose Tissue, BrownAnimalsBone DensityDisease Models, AnimalFemaleGlucoseHumansInsulinIntra-Abdominal FatLeptinLipodystrophy, Congenital GeneralizedMiceMice, KnockoutOsteosclerosisAdiponectinGlucoseInsulinLeptin

Identifiers

PMID31233501
PMCPMC6611650
OpenAlexW2953176690

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.