Bone Mineral Density Changes in Multiple Endocrine Neoplasia Type 1: A Systematic Review and Meta-Analysis of Prevalence and Parathyroidectomy Outcomes

Abstract

Objectives: This study aimed to analyze the prevalence of osteopenia and osteoporosis in MEN1-related primary hyperparathyroidism (PHPT), examine the impact of parathyroidectomy (PTX) on bone metabolic outcomes, and compare bone density metrics between sporadic and MEN1-related PHPT. Methods: A systematic review and meta-analysis were conducted in accordance with the guidelines for Meta-Analyses and Systematic Reviews of Observational Studies (MOOSE). We searched PubMed, Web of Science, and Scopus up to June 2024, subsequently screening the articles to identify relevant research. Studies focusing on bone mineral density (BMD), T and Z-scores in patients with MEN1-related conditions were included. Meta-analyses were conducted using random-effects models. Results: From the initial 2,563 articles, 15 studies were included in the meta-analysis. The pooled prevalence of osteoporosis and osteopenia in patients with MEN1-related PHPT was 45.2% (95% CI: 39.1-51.4%; I2: 16.7%) and 53.3% (95% CI: 44.4-62.0%; I2: 36.15%), respectively. PTX showed no significant impact on BMD in MEN1-related PHPT patients at the lumbar spine (mean difference: -0.054; P-value = 0.092; I2: 0.86%) or femoral neck (mean difference: -0.025; P-value = 0.219; I2: 0.47%). Comparisons of bone density metrics showed that MEN1-related PHPT patients had significantly lower Z-scores at the lumbar spine (mean difference: -0.676; P-value < 0.001; I2: 41.86%), total hip (mean difference: -0.629; P < 0.001; I2: 23.4%), and femoral neck (mean difference: -0.516; P < 0.001; I2 = 38.82%) compared to patients with sporadic PHPT. Conclusion: Patients with MEN1-related PHPT exhibited a high prevalence of osteopenia and osteoporosis, along with lower BMD metrics compared to those with sporadic PHPT. PTX was not associated with significant changes in BMD among MEN1-related PHPT patients. Level of evidence: V Objectives: This study aimed to analyze the prevalence of osteopenia and osteoporosis in MEN1-related primary hyperparathyroidism (PHPT), examine the impact of parathyroidectomy (PTX) on bone metabolic outcomes, and compare bone density metrics between sporadic and MEN1-related PHPT. Methods: A systematic review and meta-analysis were conducted in accordance with the guidelines for Meta-Analyses and Systematic Reviews of Observational Studies (MOOSE). We searched PubMed, Web of Science, and Scopus up to June 2024, subsequently screening the articles to identify relevant research. Studies focusing on bone mineral density (BMD), T and Z-scores in patients with MEN1-related conditions were included. Meta-analyses were conducted using random-effects models. Results: From the initial 2,563 articles, 15 studies were included in the meta-analysis. The pooled prevalence of osteoporosis and osteopenia in patients with MEN1-related PHPT was 45.2% (95% CI: 39.1-51.4%; I2: 16.7%) and 53.3% (95% CI: 44.4-62.0%; I2: 36.15%), respectively. PTX showed no significant impact on BMD in MEN1-related PHPT patients at the lumbar spine (mean difference: -0.054; P-value = 0.092; I2: 0.86%) or femoral neck (mean difference: -0.025; P-value = 0.219; I2: 0.47%). Comparisons of bone density metrics showed that MEN1-related PHPT patients had significantly lower Z-scores at the lumbar spine (mean difference: -0.676; P-value < 0.001; I2: 41.86%), total hip (mean difference: -0.629; P < 0.001; I2: 23.4%), and femoral neck (mean difference: -0.516; P < 0.001; I2 = 38.82%) compared to patients with sporadic PHPT. Conclusion: Patients with MEN1-related PHPT exhibited a high prevalence of osteopenia and osteoporosis, along with lower BMD metrics compared to those with sporadic PHPT. PTX was not associated with significant changes in BMD among MEN1-related PHPT patients. Level of evidence: V