Effects of Cushing disease on bone mineral density in a pediatric population

Maya B Lodish¹, Hui-Pin Hsiao², Anastasios Serbis³, Ninet Sinaii⁴, Anya Rothenbuhler³, Margaret F Keil³, Sosipatros A Boikos³, James C Reynolds⁵, Constantine A Stratakis⁶

Affiliations

¹ Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD; Pediatric Endocrinology Inter-Institute Training Program, National Institutes of Health, Bethesda, MD. Electronic address: lodishma@mail.nih.gov.
² Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD; Department of Pediatrics, Kaohsiung Municipal HsiaoKang Hospital and College of Medicine, Kaohsiung Medical University, Taiwan.
³ Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD.
⁴ Biostatistics and Clinical Epidemiology Service, National Institutes of Health Clinical Center, Bethesda, MD.
⁵ Department of Radiology, Nuclear Medicine Division, Warren Grant Magnuson Clinical Center, National Institutes of Health, Bethesda, MD.
⁶ Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD; Pediatric Endocrinology Inter-Institute Training Program, National Institutes of Health, Bethesda, MD.

PMID: 20223476
PMCID: PMC2875346
DOI: 10.1016/j.jpeds.2009.12.027

Effects of Cushing disease on bone mineral density in a pediatric population

Maya B Lodish et al. J Pediatr. 2010 Jun.

. 2010 Jun;156(6):1001-1005.

doi: 10.1016/j.jpeds.2009.12.027. Epub 2010 Mar 10.

Authors

Maya B Lodish¹, Hui-Pin Hsiao², Anastasios Serbis³, Ninet Sinaii⁴, Anya Rothenbuhler³, Margaret F Keil³, Sosipatros A Boikos³, James C Reynolds⁵, Constantine A Stratakis⁶

Affiliations

¹ Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD; Pediatric Endocrinology Inter-Institute Training Program, National Institutes of Health, Bethesda, MD. Electronic address: lodishma@mail.nih.gov.
² Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD; Department of Pediatrics, Kaohsiung Municipal HsiaoKang Hospital and College of Medicine, Kaohsiung Medical University, Taiwan.
³ Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD.
⁴ Biostatistics and Clinical Epidemiology Service, National Institutes of Health Clinical Center, Bethesda, MD.
⁵ Department of Radiology, Nuclear Medicine Division, Warren Grant Magnuson Clinical Center, National Institutes of Health, Bethesda, MD.
⁶ Section on Endocrinology Genetics, Program on Developmental Endocrinology Genetics, Eunice Kennedy Shriver National Institute of Child Health & Human Development, National Institutes of Health, Bethesda, MD; Pediatric Endocrinology Inter-Institute Training Program, National Institutes of Health, Bethesda, MD.

PMID: 20223476
PMCID: PMC2875346
DOI: 10.1016/j.jpeds.2009.12.027

Abstract

Objective: To evaluate bone mineral density (BMD) in children with Cushing disease before and after transphenoidal surgery (TSS).

Study design: Hologic dual-energy x-ray absorptiometry (DXA) scans of 35 children with Cushing disease were analyzed retrospectively. Sixteen of the 35 patients had follow-up DXA scans performed 13 to 18 months after TSS. BMD and bone mineral apparent density (BMAD) for lumbar spine (LS) L1 to L4 and femoral neck (FN) were calculated.

Results: Preoperatively, 38% and 23% of patients had osteopenia of the LS and FN, respectively. Both BMD and BMAD Z-scores of the LS were worse than those for the FN (-1.60 +/- 1.37 versus -1.04 +/- 1.19, P = .003), and (-1.90 +/- 1.49 versus -0.06 +/- 1.90, P < .001); postoperative improvement in BMD and BMAD were more pronounced in LS than in the FN (0.84 +/- 0.88 versus 0.15 +/- 0.62, P<.001; and 0.73 +/- 1.13 versus -0.26 +/- 1.21, P = .015). Pubertal stage, cortisol levels, and length of disease had no effect on BMD.

Conclusions: In children with Cushing disease, vertebral BMD was more severely affected than femoral BMD and this effect was independent of degree or duration of hypercortisolism. BMD for the LS improved significantly after TSS; osteopenia in this group may be reversible.

Published by Mosby, Inc.

PubMed Disclaimer

Conflict of interest statement

The authors declare no conflicts of interest.

Figures

**Figure 1**
A, Distributional plot for pre-operative lumbar spine and femoral neck BMD Z-scores. Dashed line is at the −2.00 level. Thick black line represents the means (−1.60 ± 1.37 vs. −1.04 ± 1.19, p=0.003). B, Distributional plot for lumbar spine BMD z-scores at baseline and follow up. Delta of 0.84 ± 0.88 (p=0.002). C, Distributional plot for femoral neck BMD z-scores at baseline and follow-up. Delta of 0.15 ± 0.62 (p=0.335).

**Figure 2**
A, Distributional plot for pre-operative lumbar spine and femoral neck BMAD Z-scores. Dashed line is at the −2.00 level. Thick black line represent the means (−1.90 ± 1.49 vs. −0.06 ± 1.90, p<.001). B, Distributional plot for lumbar spine BMAD z-scores at baseline and follow up. Delta of 0.73 ± 1.13 (p=0.021). C, Distributional plot for femoral neck BMD z-scores at baseline and follow-up. Delta of −0.26 ± 1.21 (p=0.401).

See this image and copyright information in PMC

References

1. Magiakou MA, Mastorakos G, Oldfield EH, Gomez MT, Doppman JL, Cutler GB, Jr, et al. Cushing’s syndrome in children and adolescents. Presentation, diagnosis, and therapy. N Engl J Med. 1994;331:629–636. - PubMed
1. Futo L, Toke J, Patocs A, Szappanos A, Varga I, Glaz E, et al. Skeletal differences in bone mineral area and content before and after cure of endogenous Cushing’s syndrome. Osteoporos Int. 2008;19:941–949. - PubMed
1. Godang K, Ueland T, Bollerslev J. Decreased bone area, bone mineral content, formative markers, and increased bone resorptive markers in endogenous Cushing’s syndrome. Eur J Endocrinol. 1999;141:126–131. - PubMed
1. Hermus AR, Smals AG, Swinkels LM, Huysmans DA, Pieters GF, Sweep CF, et al. Bone mineral density and bone turnover before and after surgical cure of Cushing’s syndrome. J Clin Endocrinol Metab. 1995;80:2859–2865. - PubMed
1. Kawamata A, Iihara M, Okamoto T, Obara T. Bone mineral density before and after surgical cure of Cushing’s syndrome due to adrenocortical adenoma: prospective study. World J Surg. 2008;32:890–896. - PubMed

Publication types

Actions

MeSH terms

Actions
Actions
Actions
Actions
Actions
Actions
Actions
Actions
Actions
Actions
Actions
Actions
Actions
Actions

Save citation to file

Email citation

Add to Collections

Add to My Bibliography

Your saved search

Create a file for external citation management software

Your RSS Feed

Effects of Cushing disease on bone mineral density in a pediatric population

Affiliations

Effects of Cushing disease on bone mineral density in a pediatric population

Authors

Affiliations

Abstract

Conflict of interest statement

Figures

References

Publication types

MeSH terms

Substances

Grants and funding

LinkOut - more resources

Full Text Sources

Medical

Miscellaneous