Growth characteristics in individuals with osteogenesis imperfecta in North America: results from a multicenter study.


Journal

Genetics in medicine : official journal of the American College of Medical Genetics
ISSN: 1530-0366
Titre abrégé: Genet Med
Pays: United States
ID NLM: 9815831

Informations de publication

Date de publication:
02 2019
Historique:
received: 18 10 2017
accepted: 09 04 2018
pubmed: 5 7 2018
medline: 19 4 2019
entrez: 5 7 2018
Statut: ppublish

Résumé

Osteogenesis imperfecta (OI) predisposes people to recurrent fractures, bone deformities, and short stature. There is a lack of large-scale systematic studies that have investigated growth parameters in OI. Using data from the Linked Clinical Research Centers, we compared height, growth velocity, weight, and body mass index (BMI) in 552 individuals with OI. Height, weight, and BMI were plotted on Centers for Disease Control and Prevention normative curves. In children, the median z-scores for height in OI types I, III, and IV were -0.66, -6.91, and -2.79, respectively. Growth velocity was diminished in OI types III and IV. The median z-score for weight in children with OI type III was -4.55. The median z-scores for BMI in children with OI types I, III, and IV were 0.10, 0.91, and 0.67, respectively. Generalized linear model analyses demonstrated that the height z-score was positively correlated with the severity of the OI subtype (P < 0.001), age, bisphosphonate use, and rodding (P < 0.05). From the largest cohort of individuals with OI, we provide median values for height, weight, and BMI z-scores that can aid the evaluation of overall growth in the clinic setting. This study is an important first step in the generation of OI-specific growth curves.

Identifiants

pubmed: 29970925
doi: 10.1038/s41436-018-0045-1
pii: S1098-3600(21)04612-8
pmc: PMC6320321
mid: NIHMS961139
doi:

Substances chimiques

Diphosphonates 0
Pamidronate OYY3447OMC

Types de publication

Journal Article Multicenter Study Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

275-283

Subventions

Organisme : NIGMS NIH HHS
ID : T32 GM007526
Pays : United States
Organisme : NIAMS NIH HHS
ID : U54 AR068069
Pays : United States
Organisme : NICHD NIH HHS
ID : U54 HD083092
Pays : United States
Organisme : NCATS NIH HHS
ID : UL1 TR002369
Pays : United States

Investigateurs

David R Eyre (DR)
Deborah Krakow (D)
Laura Tosi (L)
Cathleen L Raggio (CL)
Eric S Orwoll (ES)
Eric T Rush (ET)

Références

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Auteurs

Mahim Jain (M)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
Department of Bone and Osteogenesis Imperfecta, Kennedy Krieger Institute, Baltimore, Maryland, USA.

Allison Tam (A)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.

Jay R Shapiro (JR)

Department of Bone and Osteogenesis Imperfecta, Kennedy Krieger Institute, Baltimore, Maryland, USA.

Robert D Steiner (RD)

Departments of Pediatrics and Molecular and Medical Genetics, Oregon Health and Science University, Portland, Oregon, USA.
University of Wisconsin School of Medicine and Public Health, Madison, Wisconsin, USA.

Peter A Smith (PA)

Shriners Hospitals for Children, Chicago, Illinois, USA.

Michael B Bober (MB)

Division of Medical Genetics, Alfred I. duPont Hospital for Children, Wilmington, Delaware, USA.

Tracy Hart (T)

Osteogenesis Imperfecta Foundation, Gaithersburg, Maryland, USA.

David Cuthbertson (D)

College of Medicine, University of South Florida, Tampa, Florida, USA.

Jeff Krischer (J)

College of Medicine, University of South Florida, Tampa, Florida, USA.

Mary Mullins (M)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.

Sunil Bellur (S)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.

Peter H Byers (PH)

Departments of Medicine and Pathology, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.

Melanie Pepin (M)

Departments of Medicine and Pathology, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.

Michaela Durigova (M)

Shriners Hospital for Children, Canada and McGill University, Montreal, Quebec, Canada.

Francis H Glorieux (FH)

Shriners Hospital for Children, Canada and McGill University, Montreal, Quebec, Canada.

Frank Rauch (F)

Shriners Hospital for Children, Canada and McGill University, Montreal, Quebec, Canada.

Brendan Lee (B)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
Texas Children's Hospital, Houston, Texas, USA.

V Reid Sutton (VR)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
Texas Children's Hospital, Houston, Texas, USA.

Sandesh C S Nagamani (SCS)

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA. Nagamani@bcm.edu.
Texas Children's Hospital, Houston, Texas, USA. Nagamani@bcm.edu.

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