Journal
JOURNAL OF PEDIATRICS
Volume 150, Issue 1, Pages 83-88Publisher
MOSBY-ELSEVIER
DOI: 10.1016/j.jpeds.2006.10.048
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Funding
- NATIONAL CENTER FOR RESEARCH RESOURCES [M01RR000064] Funding Source: NIH RePORTER
- NATIONAL INSTITUTE OF NEUROLOGICAL DISORDERS AND STROKE [K23NS052500, R01NS050509] Funding Source: NIH RePORTER
- NCRR NIH HHS [M01-RR00064, M01 RR000064] Funding Source: Medline
- NINDS NIH HHS [1 K23 NS052500, R01 NS050509-01A1, K23 NS052500] Funding Source: Medline
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Objective To assess whether children and adolescents with neurofibromatosis type I (NF1) have decreased bone mineral density (BNID). Study design Bone densitometry of the whole body, flip, and lumbar spine was used in a case-to-control design (84 individuals with NF1:293 healthy individuals without NF1). Subjects were 5 to 18 years old. Subjects with NF1 were compared with control subjects by using an analysis-of-covariance with a fixed set of covariates (age, weight, height, Tanner stage, and sex). Results Subjects with NF1 had decreased areal BMD (aBMD) of the hip (P < .0001), femoral neck (P < .0001), lumbar spine (P = .0025). and whole body subtotal (P < .0001). When subjects with NF1 were separated in groups with and without a skeletal abnormality. those who did not have a skeletal abnormality still had statistically significant decreases in aBMD compared with control subjects (P < .0001 for whole body subtotal aBMD), although they were less pronounced than in those with osseous abnormalities. Conclusions These data suggest that individuals with NF1 have a unique generalized skeletal dysplasia, predisposing them to localized osseous defects. Dual energy x-ray absorptiometry may prove useful in identifying individuals with NF1 who are at risk for clinical osseous complications and monitoring therapeutic trials.
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