Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis

Abstract Introduction Patients with haemophilia (PWH) may have lower bone mineral density (BMD). The risk of low BMD in PWH has not been comprehensively analysed. This study aimed to examine the risk of low BMD and changes in BMD in PWH. Methods A comprehensive systematic search was performed in 4 d...

Full description

Bibliographic Details
Main Authors: Haojing Zhou, Lei Chen, Hai Su, Guoqian Chen, Peijian Tong
Format: Article
Language:English
Published: BMC 2024-01-01
Series:Journal of Orthopaedic Surgery and Research
Subjects:
Online Access:https://doi.org/10.1186/s13018-023-04499-6
_version_ 1827382049590214656
author Haojing Zhou
Lei Chen
Hai Su
Guoqian Chen
Peijian Tong
author_facet Haojing Zhou
Lei Chen
Hai Su
Guoqian Chen
Peijian Tong
author_sort Haojing Zhou
collection DOAJ
description Abstract Introduction Patients with haemophilia (PWH) may have lower bone mineral density (BMD). The risk of low BMD in PWH has not been comprehensively analysed. This study aimed to examine the risk of low BMD and changes in BMD in PWH. Methods A comprehensive systematic search was performed in 4 databases: PubMed, Embase, Web of Science, and Cochrane Library. The last search was carried out on 11 December 2022. Review Manager 5.4 and Stata 16 were used for meta-analysis. Odds ratios were calculated by the incidence of low BMD between the haemophilia and control groups in each study. A meta-analysis of the odds ratios for each study was performed to estimate pooled odds ratios. Fixed effects models or random effects models were used to assess outcomes. Heterogeneity was evaluated using Higgins’ I2. Subgroup analysis and sensitivity analysis were performed to interpret the potential source of heterogeneity. A funnel plot, Egger’s regression test, and the trim-and-fill method were used to assess publication bias. Results 19 of 793 studies, published between 2004 and 2022, that were identified by search strategy were included in this meta-analysis. The risk for low BMD was approximately four times higher compared to controls. PWH have significantly lower lumbar spine, femoral neck, and total hip BMD. Subgroup analysis showed that the risk of low BMD did not increase significantly in developed countries. Very low heterogeneity was observed in the meta-analysis of the risk of low BMD. The result from Egger’s regression test suggested that there may be publication bias. However, the meta-analysis results did not alter after the trim-and-fill correction and the findings were robust. Conclusion Haemophilia was associated with an increased risk of low BMD. However, the risk of low BMD did not increase significantly in developed countries. And BMD was reduced in PWH, regardless of age, region, or economic ability. For PWH, our concerns should extend beyond bleeding and osteoarthritis to encompass BMD starting at a young age.
first_indexed 2024-03-08T14:13:53Z
format Article
id doaj.art-2875c5d200a34eeaa5f931e4834870c9
institution Directory Open Access Journal
issn 1749-799X
language English
last_indexed 2024-03-08T14:13:53Z
publishDate 2024-01-01
publisher BMC
record_format Article
series Journal of Orthopaedic Surgery and Research
spelling doaj.art-2875c5d200a34eeaa5f931e4834870c92024-01-14T12:30:09ZengBMCJournal of Orthopaedic Surgery and Research1749-799X2024-01-0119111210.1186/s13018-023-04499-6Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysisHaojing Zhou0Lei Chen1Hai Su2Guoqian Chen3Peijian Tong4Zhejiang Chinese Medical UniversityZhejiang Chinese Medical UniversityZhejiang Chinese Medical UniversityThe First Affiliated Hospital of Zhejiang Chinese Medical University (Zhejiang Provincial Hospital of Chinese Medicine)The First Affiliated Hospital of Zhejiang Chinese Medical University (Zhejiang Provincial Hospital of Chinese Medicine)Abstract Introduction Patients with haemophilia (PWH) may have lower bone mineral density (BMD). The risk of low BMD in PWH has not been comprehensively analysed. This study aimed to examine the risk of low BMD and changes in BMD in PWH. Methods A comprehensive systematic search was performed in 4 databases: PubMed, Embase, Web of Science, and Cochrane Library. The last search was carried out on 11 December 2022. Review Manager 5.4 and Stata 16 were used for meta-analysis. Odds ratios were calculated by the incidence of low BMD between the haemophilia and control groups in each study. A meta-analysis of the odds ratios for each study was performed to estimate pooled odds ratios. Fixed effects models or random effects models were used to assess outcomes. Heterogeneity was evaluated using Higgins’ I2. Subgroup analysis and sensitivity analysis were performed to interpret the potential source of heterogeneity. A funnel plot, Egger’s regression test, and the trim-and-fill method were used to assess publication bias. Results 19 of 793 studies, published between 2004 and 2022, that were identified by search strategy were included in this meta-analysis. The risk for low BMD was approximately four times higher compared to controls. PWH have significantly lower lumbar spine, femoral neck, and total hip BMD. Subgroup analysis showed that the risk of low BMD did not increase significantly in developed countries. Very low heterogeneity was observed in the meta-analysis of the risk of low BMD. The result from Egger’s regression test suggested that there may be publication bias. However, the meta-analysis results did not alter after the trim-and-fill correction and the findings were robust. Conclusion Haemophilia was associated with an increased risk of low BMD. However, the risk of low BMD did not increase significantly in developed countries. And BMD was reduced in PWH, regardless of age, region, or economic ability. For PWH, our concerns should extend beyond bleeding and osteoarthritis to encompass BMD starting at a young age.https://doi.org/10.1186/s13018-023-04499-6Bone densityHaemophiliaMeta-analysisOsteopeniaOsteoporosis
spellingShingle Haojing Zhou
Lei Chen
Hai Su
Guoqian Chen
Peijian Tong
Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis
Journal of Orthopaedic Surgery and Research
Bone density
Haemophilia
Meta-analysis
Osteopenia
Osteoporosis
title Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis
title_full Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis
title_fullStr Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis
title_full_unstemmed Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis
title_short Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis
title_sort risk of low bone mineral density in patients with haemophilia a systematic review and meta analysis
topic Bone density
Haemophilia
Meta-analysis
Osteopenia
Osteoporosis
url https://doi.org/10.1186/s13018-023-04499-6
work_keys_str_mv AT haojingzhou riskoflowbonemineraldensityinpatientswithhaemophiliaasystematicreviewandmetaanalysis
AT leichen riskoflowbonemineraldensityinpatientswithhaemophiliaasystematicreviewandmetaanalysis
AT haisu riskoflowbonemineraldensityinpatientswithhaemophiliaasystematicreviewandmetaanalysis
AT guoqianchen riskoflowbonemineraldensityinpatientswithhaemophiliaasystematicreviewandmetaanalysis
AT peijiantong riskoflowbonemineraldensityinpatientswithhaemophiliaasystematicreviewandmetaanalysis