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Licensed Unlicensed Requires Authentication Published by De Gruyter August 3, 2023

Longitudinal assessment of bone health index as a measure of bone health in short-statured children before and during treatment with recombinant growth hormone

  • Lukas Holzapfel EMAIL logo , Daniela Choukair , Jens-Peter Schenk and Markus Bettendorf EMAIL logo

Abstract

Objectives

The aim of our study was the longitudinal assessment of bone health index (BHI) in short-statured children during growth hormone (GH) treatment to estimate changes in their bone health.

Methods

256 short-statured children (isolated GH deficiency (IGHD) n=121, multiple pituitary hormone deficiency (MPHD) n=49, intrauterine growth retardation (small for gestational age (SGA)) n=52, SHOX (short stature homeobox gene) deficiency n=9, Ullrich Turner syndrome (UTS) n=25) who started with GH between 2010 and 2018 were included. Annual bone ages (Greulich and Pyle, GP) and BHI were, retrospectively, analysed in consecutive radiographs of the left hand (BoneXpert software) from GH therapy start (T0) up to 10 years (T10) thereafter, with T max indicating the individual time point of the last available radiograph. The results are presented as the median (25 %/75 % interquartile ranges, IQR) and statistical analyses were performed using non-parametric tests as appropriate.

Results

The BHI standard deviation scores (SDS) were reduced (−0.97, −1.8/−0.3) as bone ages were retarded (−1.6 years, −2.31/−0.97) in all patients before start of GH and were significantly lower in patients with growth hormone deficiency (GHD) (−1.04, −1.85/−0.56; n=170) compared to non-GHD patients (−0.79, −1.56/−0.01; n=86; p=0.022). BHI SDS increased to −0.17 (−1/0.58) after 1 year of GH (T1, 0.5–1.49, p<0.001) and to −0.20 (−1/−0.50, p<0.001) after 5.3 years (T max, 3.45/7.25).

Conclusions

BHI SDS are reduced in treatment-naive short-statured children regardless of their GH status, increase initially with GH treatment while plateauing thereafter, suggesting sustained improved bone health.


Corresponding authors: Lukas Holzapfel and Prof. Dr. med. Markus Bettendorf, Division of Paediatric Endocrinology and Diabetes, Department of Paediatrics, University Hospital Heidelberg, Im Neuenheimer Feld 430 69120 Heidelberg, Germany, Phone: 0176/43846735, E-mail: Phone: 06221 564002, E-mail: (M. Bettendorf)
Presented in part at the 60th Annual meeting of the European Society of Paediatric Endocrinology 2022 in Rome, Italy.
  1. Research funding: None declared.

  2. Author contributions: All authors have accepted responsibility for the entire content of this manuscript and approved its submission. LH, DC and MB planned the study, wrote study protocol, conducted study and collected data. LH evaluated the data, performed statistical analyses and created the graphs. LH, DC, MR, JPS and MB wrote the manuscript and interpreted the final results. All authors performed proof reading of the final version of this manuscript. All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.

  3. Competing interests: Authors state no competing of interest.

  4. Informed consent: Not applicable. The Ethics Committee of University Hospital Heidelberg approved the study (S-029/2019) and decided that written consents of study participants were not required. The identification of personal information was not necessary for scientific reasons. All data were used pseudonymized.

  5. Ethical approval: The Ethics Committee of University Hospital Heidelberg approved the study (S-029/2019). Written consents of study participants were not required. Study was conducted according to the recommendations of the Declaration of Helsinki.

  6. Data availability: The data is available from the corresponding author (LH) upon reasonable request.

  7. Definition of research data: Primary data.

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Received: 2023-02-27
Accepted: 2023-07-11
Published Online: 2023-08-03
Published in Print: 2023-09-26

© 2023 Walter de Gruyter GmbH, Berlin/Boston

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