Open Patient Data Explorative Network (OPEN), Department of Clinical Research, University of Southern Denmark and Odense University Hospital, 5000, Odense C, Denmark.
Department of Medicine, Holbæk Hospital, 4300, Holbæk, Denmark.
Calcif Tissue Int. 2021 Aug;109(2):147-156. doi: 10.1007/s00223-021-00843-2. Epub 2021 Apr 5.
Tumor-induced osteomalacia (TIO) is a rare, acquired condition of phosphate wasting due to phosphaturic mesenchymal tumors. Because the incidence and prevalence of TIO is unknown, we conducted an observational cohort study using national Danish health registers for the period 2008 to 2018 to obtain such information. The study also aimed to describe the demographics of the TIO population and the prognosis. The operational definition was based on hypophosphatemia or adult osteomalacia diagnoses, combined with prescriptions used in the initial management and procedures consistent with advanced imaging used for locating tumors. The incidence of TIO in Denmark was found to be below 0.13 per 100,000 person years for the total population of the country and 0.10 per 100,000 in adult-onset disease. The prevalence of TIO was estimated to be no more than 0.70 per 100,000 persons for the total population and 0.43 per 100,000 in adults. In 2018, there were a maximum of nine new cases of TIO in Danish adults. Mortality was low but few patients fulfilled the protocol cure criterion during the observation period. TIO has no ICD-10 code and limitations to the study include lack of information on serum biochemistry and on the use of phosphate supplements. Strengths include the use of long-term longitudinal, national hospital and prescription data from a country with universal healthcare. Given the very small patient population with TIO and the known delay to diagnosis and cure, management of patients with suspected TIO should be centralized.
肿瘤相关性骨软化症(TIO)是一种罕见的获得性磷酸盐丢失疾病,由致磷酸盐尿的间叶性肿瘤引起。由于 TIO 的发病率和患病率未知,我们开展了一项观察性队列研究,使用丹麦全国卫生登记系统,对 2008 年至 2018 年的数据进行分析,以获得相关信息。该研究还旨在描述 TIO 患者的人口统计学特征和预后。其操作性定义基于低磷血症或成人骨软化症的诊断,同时结合初始管理中的处方以及用于定位肿瘤的先进影像学检查方法。丹麦 TIO 的总发病率为每 10 万人年低于 0.13 例,成人发病者为每 10 万人年 0.10 例。TIO 的总患病率估计不超过每 10 万人 0.70 例,成人的患病率为每 10 万人 0.43 例。2018 年,丹麦成年 TIO 患者最多有 9 例新发病例。死亡率较低,但在观察期间,很少有患者符合方案治愈标准。TIO 没有 ICD-10 编码,该研究的局限性包括缺乏血清生化和磷酸盐补充剂使用情况的信息。其优势在于使用了长期的纵向、全国性的医院和处方数据,且来自一个全民医保的国家。鉴于 TIO 患者的人数非常少,且已知诊断和治愈存在延迟,疑似 TIO 患者的管理应集中进行。