Abstract
Background
Determination of bone age is routinely used for following up substitution therapy in congenital adrenal hyperplasia (CAH) but today is a procedure with significant subjectivity.
Objective
The aim was to test the performance of automatic bone age rating by the BoneXpert software package in all radiographs of children with CAH seen at our clinic from 1975 to 2006.
Materials and methods
Eight hundred and ninety-two left-hand radiographs from 100 children aged 0 to 17 years were presented to a human rater and BoneXpert for bone age rating. Images where ratings differed by more than 1.5 years were each rerated by four human raters.
Results
Rerating was necessary in 20 images and the rerating result was closer to the BoneXpert result than to the original manual rating in 18/20 (90 %). Bone age rating precision based on the smoothness of longitudinal curves comprising a total of 327 data triplets spanning less than 1.7 years showed BoneXpert to be more precise (P<0.001).
Conclusion
BoneXpert performs reliable bone age ratings in children with CAH.
Similar content being viewed by others
Notes
The triplets formed from a child are not statistically independent because each visit can participate in up to three triplets. The confidence interval (CI) of the estimated precision was therefore estimated by a Monte Carlo technique, which showed that the CIs are a factor of 1.3 times larger than one would derive by assuming that the triplets were independent
References
White PC, Speiser PW (2000) Congenital adrenal hyperplasia due to 21-hydroxylase deficiency. Endocr Rev 21:245–291
Koletzko B (2007) Kinder-und Jugendmedizin, Heidelberg: Springer Medizin Verlag, Heidelberg
Pang SY, Wallace MA, Hofman L et al (1998) Worldwide experience in newborn screening for classical congenital adrenal hyperplasia due to 21-hydroxylase deficiency. Pediatrics 81:866–874
David M, Sempe M, Blanc M et al (1994) Final height in 69 patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency. Arch Pediatr 1:363–367
Ghali I, David M, David L (1978) Linear growth and pubertal development in treated congenital adrenal hyperplasia due to 21-hydroxylase deficiency. Obstet Gynecol Surv 33:120–122
Jääskeläinen J, Voutilainen R (1997) Growth of patients with 21-hydroxylase deficiency: an analysis of the factors influencing adult height. Pediatr Res 41:30–33
Muirhead S, Sellers EAC, Guyda H (2002) Indicators of adult height outcome in classical 21-hydroxylase deficiency congenital adrenal hyperplasia. J Pediatr 141:247–252
Van der Kamp HJ, Otten BJ, Buitenweg N et al (2002) Longitudinal analysis of growth and puberty in 21-hydroxylase deficiency patients. Arch Dis Child 87:139–144
Bonfig W, Bechtold S, Schmidt H et al (2007) Reduced final height outcome in congenital adrenal hyperplasia under prednisone treatment: deceleration of growth velocity during puberty. J Clin Endocrinol Metabol 92:1635–1639
Eugster EA, DiMeglio LA, Wright JC et al (2001) Height outcome in congenital adrenal hyperplasia caused by 21-hydroxylase deficiency: a meta-analysis. J Pediatr 138:26–32
Hargitai G, Solyom J, Battelino T et al (2000) Growth patterns and final height in congenital adrenal hyperplasia due to classical 21-hydroxylase deficiency. Horm Res 55:161–171
Lin-Su K, Vogiatzi MG, Marshall I et al (2005) Treatment with growth hormone and luteinizing hormone releasing hormone analog improves final adult height in children with congenital adrenal hyperplasia. J Clin Endocrinol Metabol 90:3318–3325
Martin DD, Neuhof J, Jenni OG et al (2010) Automatic determination of left-and right-hand bone age in the First Zurich Longitudinal Study. Horm Res Paediatr 74:50–55
Thodberg HH, Jenni OG, Caflisch J et al (2009) Prediction of adult height based on automated determination of bone age. J Clin Endocrinol Metab 94:4868–4874
Thodberg HH, Neuhof J, Ranke MB et al (2010) Validation of bone age methods by their ability to predict adult height. Horm Res Paediatr 74:15–22
van Rijn RR, Lequin MH, Thodberg HH (2009) Automatic determination of Greulich and Pyle bone age in healthy Dutch children. Pediatr Radiol 39:591–597
Martin DD, Deusch D, Schweizer R et al (2009) Clinical application of automated Greulich-Pyle bone age determination in children with short stature. Pediatr Radiol 39:598–607
Martin DD, Meister K, Schweizer R et al (2008) Validation of automatic bone age rating in children with precocious and early puberty. J Pediatr Endocrinol Metab. 2011;24(11-12):1009–1014
Thodberg HH, Kreiborg S, Juul A et al (2009) The BoneXpert method for automated determination of skeletal maturity. IEEE Trans Med Imaging 28:52–66
Berst MJ, Dolan L, Bogdanowicz MM et al (2001) Effect of knowledge of chronologic age on the variability of pediatric bone age determined using the Greulich and Pyle standards. AJR Am J Roentgonol 176:507–510
King DG, Steventon DM, O’Sullivan MP et al (1994) Reproducibility of bone ages when performed by radiology registrars: an audit of Tanner and Whitehouse II versus Greulich and Pyle methods. Br J Radiol 67:848–851
Conflicts of interest
None.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Martin, D.D., Heil, K., Heckmann, C. et al. Validation of automatic bone age determination in children with congenital adrenal hyperplasia. Pediatr Radiol 43, 1615–1621 (2013). https://doi.org/10.1007/s00247-013-2744-8
Received:
Revised:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00247-013-2744-8