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

, Members Of The Brittle Bone Disorders Consortium*,

Research output: Contribution to journalArticle

1 Citation (Scopus)

Abstract

Purpose: 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. Methods: 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. Results: 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). Conclusion: 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.

Original languageEnglish (US)
Pages (from-to)1-9
Number of pages9
JournalGenetics in Medicine
DOIs
StateAccepted/In press - Jul 4 2018

Fingerprint

Osteogenesis Imperfecta
North America
Multicenter Studies
Growth
Body Mass Index
Weights and Measures
Bone Fractures
Diphosphonates
Centers for Disease Control and Prevention (U.S.)
Linear Models
Type 3 Osteogenesis imperfecta

ASJC Scopus subject areas

  • Genetics(clinical)

Cite this

Growth characteristics in individuals with osteogenesis imperfecta in North America : results from a multicenter study. / , Members Of The Brittle Bone Disorders Consortium*,.

In: Genetics in Medicine, 04.07.2018, p. 1-9.

Research output: Contribution to journalArticle

@article{5ddf1529e379498bb507e0d2331c8daa,
title = "Growth characteristics in individuals with osteogenesis imperfecta in North America: results from a multicenter study",
abstract = "Purpose: 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. Methods: 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. Results: 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). Conclusion: 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.",
author = "{, Members Of The Brittle Bone Disorders Consortium*,} and Mahim Jain and Allison Tam and Shapiro, {Jay R.} and Steiner, {Robert D.} and Smith, {Peter A.} and Bober, {Michael B.} and Tracy Hart and David Cuthbertson and Jeff Krischer and Mary Mullins and Sunil Bellur and Byers, {Peter H.} and Melanie Pepin and Michaela Durigova and Glorieux, {Francis H.} and Frank Rauch and Brendan Lee and Sutton, {V. Reid} and Eyre, {David R.} and Deborah Krakow and Laura Tosi and Raggio, {Cathleen L.} and Eric Orwoll and Rush, {Eric T.} and Nagamani, {Sandesh C.S.}",
year = "2018",
month = "7",
day = "4",
doi = "10.1038/s41436-018-0045-1",
language = "English (US)",
pages = "1--9",
journal = "Genetics in Medicine",
issn = "1098-3600",
publisher = "Lippincott Williams and Wilkins",

}

TY - JOUR

T1 - Growth characteristics in individuals with osteogenesis imperfecta in North America

T2 - results from a multicenter study

AU - , Members Of The Brittle Bone Disorders Consortium,

AU - Jain, Mahim

AU - Tam, Allison

AU - Shapiro, Jay R.

AU - Steiner, Robert D.

AU - Smith, Peter A.

AU - Bober, Michael B.

AU - Hart, Tracy

AU - Cuthbertson, David

AU - Krischer, Jeff

AU - Mullins, Mary

AU - Bellur, Sunil

AU - Byers, Peter H.

AU - Pepin, Melanie

AU - Durigova, Michaela

AU - Glorieux, Francis H.

AU - Rauch, Frank

AU - Lee, Brendan

AU - Sutton, V. Reid

AU - Eyre, David R.

AU - Krakow, Deborah

AU - Tosi, Laura

AU - Raggio, Cathleen L.

AU - Orwoll, Eric

AU - Rush, Eric T.

AU - Nagamani, Sandesh C.S.

PY - 2018/7/4

Y1 - 2018/7/4

N2 - Purpose: 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. Methods: 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. Results: 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). Conclusion: 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.

AB - Purpose: 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. Methods: 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. Results: 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). Conclusion: 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.

UR - http://www.scopus.com/inward/record.url?scp=85049515167&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85049515167&partnerID=8YFLogxK

U2 - 10.1038/s41436-018-0045-1

DO - 10.1038/s41436-018-0045-1

M3 - Article

C2 - 29970925

AN - SCOPUS:85049515167

SP - 1

EP - 9

JO - Genetics in Medicine

JF - Genetics in Medicine

SN - 1098-3600

ER -