TY - JOUR
T1 - Growth characteristics in individuals with osteogenesis imperfecta in North America
T2 - results from a multicenter study
AU - 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 S.
AU - Rush, Eric T.
AU - Nagamani, Sandesh C.S.
N1 - Publisher Copyright:
© 2018, American College of Medical Genetics and Genomics.
PY - 2019/2/1
Y1 - 2019/2/1
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.
KW - Growth
KW - Height
KW - Natural history study
KW - Osteogenesis imperfecta
KW - Weight
UR - https://www.scopus.com/pages/publications/85049515167
U2 - 10.1038/s41436-018-0045-1
DO - 10.1038/s41436-018-0045-1
M3 - Article
C2 - 29970925
AN - SCOPUS:85049515167
SN - 1098-3600
VL - 21
SP - 275
EP - 283
JO - Genetics in Medicine
JF - Genetics in Medicine
IS - 2
ER -