{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,28]],"date-time":"2026-08-28T14:24:31Z","timestamp":1787927071161,"version":"build-2784847793"},"reference-count":15,"publisher":"The Endocrine Society","license":[{"start":{"date-parts":[[2026,8,13]],"date-time":"2026-08-13T00:00:00Z","timestamp":1786579200000},"content-version":"am","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>Current tools to evaluate fracture risk in children with osteogenesis imperfecta (OI) are limited, and bone mineral density (BMD) has limitations and is not widely available for children under 5 years. We hypothesized that the Bone Health Index (BHI), evaluating the average cortical thickness of the 3 middle metacarpal bones, adjusted for bone width and length, could be a useful tool in the assessment of fracture risk, particularly in infants.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>The service at Great Ormond Street Hospital (London, United Kingdom) follows a cohort of over 277 children with classical OI and keeps a detailed and accurate record of confirmed fractures. We reviewed their BHI (analyzed using the latest version of BoneXpert\u00ae software) at presentation (n = 123), the lumbar BMD (n = 47) taken on the same day where available, the subsequent 2-year fracture history, and annual lateral spine X-rays.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>In multivariable logistic regression, each 1-SD decrease in BHI SD score (SDS) was independently associated with a 5.3-fold higher odds of incident fracture within 2 years (odds ratio 5.26, 95% CI 2.4-11.1, P &amp;lt; .001), while dual-energy X-ray absorptiometry (DXA) BMD and bone mineral apparent density (BMAD) Z-scores were not independent predictors. Receiver operating characteristic analysis showed good discriminatory performance for BHI SDS (area under the curve 0.84), superior to DXA BMD Z-score (AUC 0.60) and BMAD Z-score (AUC 0.51).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Our study indicates that lower BHI SDS values are associated with increased fracture risk in children with classical OI. In the available DXA subgroup, BHI demonstrated stronger discriminatory performance than DXA-derived measures. Bone Health Index may represent a clinically useful complementary tool for fracture risk stratification in young children with OI, particularly where DXA is unavailable or technically limited, including infancy.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1210\/clinem\/dgag336","type":"journal-article","created":{"date-parts":[[2026,8,12]],"date-time":"2026-08-12T11:51:22Z","timestamp":1786535482000},"source":"Crossref","is-referenced-by-count":0,"title":["Bone Health Index: a fracture risk assessment tool in children with osteogenesis imperfecta"],"prefix":"10.1210","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-5378-1895","authenticated-orcid":false,"given":"Ruggero","family":"Lanzafame","sequence":"first","affiliation":[{"name":"Paediatric Endocrinology Department, Great Ormond Street Hospital for Children NHS Foundation Trust , London WC1N 3JH ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alistair D","family":"Calder","sequence":"additional","affiliation":[{"name":"Radiology Department, Great Ormond Street Hospital for Children NHS Foundation Trust , London WC1N 3JH ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Belinda","family":"Crowe","sequence":"additional","affiliation":[{"name":"Neurosciences Department, Great Ormond Street Hospital for Children NHS Foundation Trust , London WC1N 3JH ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Catherine","family":"DeVile","sequence":"additional","affiliation":[{"name":"Neurosciences Department, Great Ormond Street Hospital for Children NHS Foundation Trust , London WC1N 3JH ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0742-7595","authenticated-orcid":false,"given":"Moira S","family":"Cheung","sequence":"additional","affiliation":[{"name":"Paediatric Endocrinology Department, Great Ormond Street Hospital for Children NHS Foundation Trust , London WC1N 3JH ,","place":["UK"]},{"name":"NIHR Great Ormond Street Hospital Biomedical Research Centre, University College London , London WC1N 1EH ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2026,8,13]]},"reference":[{"issue":"1","key":"2026082809212532500_dgag336-B1","doi-asserted-by":"crossref","first-page":"17052","DOI":"10.1038\/nrdp.2017.52","article-title":"Osteogenesis imperfecta","volume":"3","author":"Marini","year":"2017","journal-title":"Nat Rev Dis Primers"},{"issue":"6","key":"2026082809212532500_dgag336-B2","doi-asserted-by":"crossref","first-page":"891","DOI":"10.1007\/s00223-024-01266-5","article-title":"Update on the genetics of osteogenesis imperfecta","volume":"115","author":"Jovanovic","year":"2024","journal-title":"Calcif Tissue Int"},{"issue":"1","key":"2026082809212532500_dgag336-B3","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1021\/acsptsci.3c00324","article-title":"Emerging landscape of osteogenesis imperfecta pathogenesis and therapeutic approaches","volume":"7","author":"Sun","year":"2024","journal-title":"ACS Pharmacol Transl Sci"},{"key":"2026082809212532500_dgag336-B4","doi-asserted-by":"crossref","DOI":"10.1016\/j.bone.2021.116013","article-title":"Which skeletal imaging modality is best for assessing bone health in children and young adults compared to DXA? 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