Suppr超能文献

肿瘤诱导性骨软化症患者的骨骼如何恢复?一项全国队列研究的长期随访

How does bone recover in patients with tumor-induced osteomalacia? Long-term follow-up in a national cohort study.

作者信息

Zanchetta María Belén, Jerkovich Fernando, Scioscia Florencia, Mocarbel Yamile, Pignatta Analía, Elías Natalia, Roganovich Juan Manuel, Vigovich Carlos, Balonga María Celeste, Cohen Ana Carolina, Mumbach Giselle, Mansur José Luis, Fux Otta Carolina, Douthat Walter Guillermo, Tartaglia Pilar, Cecchi Griselda, Bastianello María, Plantalech Luisa, Fradinger Erich, Zanchetta José Rubén

机构信息

IDIM, Universidad del Salvador, Instituto de DiagnÓstico e Investigaciones MetabÓlicas, Buenos Aires, C1012AAR, Argentina.

División Endocrinología, Hospital de Clínicas, Universidad de Buenos Aires, Buenos Aires, C1120, Argentina.

出版信息

JBMR Plus. 2025 Apr 14;9(5):ziaf041. doi: 10.1093/jbmrpl/ziaf041. eCollection 2025 May.

Abstract

Tumor-induced osteomalacia (TIO) is a rare disorder characterized by impaired bone mineralization due to phosphate wasting. Long-term changes in BMD and microarchitecture after surgical cure or medical therapy in TIO are not well understood. This study describes changes in BMD, microarchitecture, and bone strength in patients with TIO following surgical cure or medical therapy. A prospective cohort study included adults diagnosed with TIO from May 2018 to 2024, categorized into those with surgical cure and those on medical therapy. Follow-up assessments were classified as early (median 8 mo), intermediate (median 17 mo), and long-term (median 26 mo). Fifteen patients were included: seven achieved surgical cure, and eight remained on medical therapy. Lumbar spine BMD increased by +19% at early, +27% at intermediate, and +15% at long-term follow-up. Total hip BMD increased by +31%, +36%, and +31% at early, intermediate, and long-term assessments, respectively. All patients achieved a normal lumbar spine BMD, while 91% attained a normal total hip BMD. At the distal tibia, substantial increases in bone microarchitecture parameters-cortical area (Ct.Ar), cortical volumetric density (Ct.vBMD), and cortical thickness (Ct.Th)-were observed. Notably, Ct.Th improved to levels comparable to healthy controls. Bone strength improved by 13% but was not statistically significant, probably due to the small sample size. At the distal radius, most parameters remained stable. Patients with surgical cure showed more rapid and substantial improvements in BMD and cortical microarchitecture than non-cured patients, but these differences did not reach statistical significance. Overall, bone recovery in TIO is gradual, with gains in spine and hip BMD and significant improvements in tibial cortical parameters. However, some aspects of bone microarchitecture remained below control levels, underscoring the need for ongoing monitoring and individualized management strategies.

摘要

肿瘤诱导的骨软化症(TIO)是一种罕见的疾病,其特征是由于磷酸盐流失导致骨矿化受损。TIO患者在手术治愈或药物治疗后骨密度(BMD)和微观结构的长期变化尚不清楚。本研究描述了TIO患者在手术治愈或药物治疗后BMD、微观结构和骨强度的变化。一项前瞻性队列研究纳入了2018年5月至2024年诊断为TIO的成年人,分为手术治愈组和药物治疗组。随访评估分为早期(中位数8个月)、中期(中位数17个月)和长期(中位数26个月)。共纳入15例患者:7例实现手术治愈,8例继续接受药物治疗。腰椎BMD在早期随访时增加了19%,中期增加了27%,长期随访时增加了15%。全髋BMD在早期、中期和长期评估时分别增加了31%、36%和31%。所有患者的腰椎BMD均恢复正常,而91%的患者全髋BMD达到正常。在胫骨远端,观察到骨微观结构参数——皮质面积(Ct.Ar)、皮质体积密度(Ct.vBMD)和皮质厚度(Ct.Th)有显著增加。值得注意的是,Ct.Th改善到与健康对照相当的水平。骨强度提高了13%但无统计学意义,可能是由于样本量小。在桡骨远端,大多数参数保持稳定。手术治愈的患者在BMD和皮质微观结构方面的改善比未治愈的患者更快、更显著,但这些差异未达到统计学意义。总体而言,TIO患者的骨恢复是渐进的,脊柱和髋部BMD增加,胫骨皮质参数有显著改善。然而,骨微观结构的某些方面仍低于对照水平,这突出了持续监测和个体化管理策略的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9d9a/12010155/7e3de2cc8c65/ziaf041f1.jpg

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验