Hai Wang1, Dingrong Zhong1, Yong Liu1, Yan Jiang1, Guixing Qiu1, Xisheng Weng1, Xiaoping Xing1, Mei Li1, Xunwu Meng1, Fang Li1, Zhaohui Zhu1, Wei Yu1, Weibo Xia1, Jin Jin1. 1. Departments of Orthopaedic Surgery (H.W., Y.L., G.Q., X.W., and J.J.), Endocrinology (Y.J., X.X., M.L., X.M., and W.X.), Pathology (D.Z.), Nuclear Medicine (F.L. and Z.Z.), and Radiology (W.Y.), Peking Union Medical College Hospital (PUMCH), Number 1 Shuaifuyuan hutong, Beijing 100730, People's Republic of China. E-mail address for H. Wang: wanghaip0386@pumch.cn. E-mail address for D. Zhong: zhongdingrong@sina.com. E-mail address for Y. Liu: liuyongxh@163.com. E-mail address for Y. Jiang: sinojenny@126.com E-mail address for G. Qiu: qiugx@medmail.com.cn. E-mail address for X. Weng: xshweng@medmail.com.cn. E-mail address for X. Xing: xingxp2006@126.com. E-mail address for M. Li: limeilzh@sina.com. E-mail address for X. Meng: mengxunwu@sina.com. E-mail address for F. Li: lifang@pumch.cn. E-mail address for Z. Zhu: zhuzhh@pumch.cn. E-mail address for W. Yu: weiyu5508@yahoo.com. E-mail address for W. Xia: xiaweibo@medmail.com.cn. E-mail address for J. Jin: jinjin@pumch.cn.
Abstract
BACKGROUND: Tumor-induced osteomalacia is a rare and fascinating paraneoplastic syndrome usually caused by a small, benign phosphaturic mesenchymal tumor. Most tumors are treated surgically, but we are unaware of any reports that compare the results of curettage and segmental resection for lesions in long bones. METHODS: Seventeen patients (ten male and seven female) with tumor-induced osteomalacia lesions in long bones, who underwent surgical treatment from December 2004 to August 2013 in our hospital, were included in this retrospective study. The mean follow-up (and standard deviation) was 35 ± 27 months (range, twelve to 116 months). The characteristics of the tumor and the effects of different surgical treatments (curettage compared with segmental resection) were evaluated. RESULTS: All patients showed typical clinical characteristics of tumor-induced osteomalacia, including elevated serum fibroblast growth factor-23 (FGF-23); 82% of tumors were in the epiphysis, and 82% grew eccentrically. The mean maximum diameter of the tumors was 2.4 ± 2.0 cm. The complete resection rates were similar for curettage (67%) and segmental resection (80%). However, the recurrence rate after curettage (50%) was higher than that after segmental resection (0%). The complete resection rate for secondary segmental resection (75%) was not different from that for primary segmental resection (83%). All of our cases of tumor-induced osteomalacia were caused by phosphaturic mesenchymal tumors. After successful removal of tumors, serum FGF-23 returned to normal within twenty-four hours and serum phosphorus levels returned to normal at a mean of 6.5 ± 3.5 days. CONCLUSIONS: Most lesions in long bones are located in the epiphysis, so curettage is first suggested to maintain joint function. If curettage is incomplete or there is a recurrence, secondary segmental resection should be considered curative. Changes of serum FGF-23 and phosphorus levels before and after the operation may be of prognostic help.
BACKGROUND:Tumor-induced osteomalacia is a rare and fascinating paraneoplastic syndrome usually caused by a small, benign phosphaturic mesenchymal tumor. Most tumors are treated surgically, but we are unaware of any reports that compare the results of curettage and segmental resection for lesions in long bones. METHODS: Seventeen patients (ten male and seven female) with tumor-induced osteomalacia lesions in long bones, who underwent surgical treatment from December 2004 to August 2013 in our hospital, were included in this retrospective study. The mean follow-up (and standard deviation) was 35 ± 27 months (range, twelve to 116 months). The characteristics of the tumor and the effects of different surgical treatments (curettage compared with segmental resection) were evaluated. RESULTS: All patients showed typical clinical characteristics of tumor-induced osteomalacia, including elevated serum fibroblast growth factor-23 (FGF-23); 82% of tumors were in the epiphysis, and 82% grew eccentrically. The mean maximum diameter of the tumors was 2.4 ± 2.0 cm. The complete resection rates were similar for curettage (67%) and segmental resection (80%). However, the recurrence rate after curettage (50%) was higher than that after segmental resection (0%). The complete resection rate for secondary segmental resection (75%) was not different from that for primary segmental resection (83%). All of our cases of tumor-induced osteomalacia were caused by phosphaturic mesenchymal tumors. After successful removal of tumors, serum FGF-23 returned to normal within twenty-four hours and serum phosphorus levels returned to normal at a mean of 6.5 ± 3.5 days. CONCLUSIONS: Most lesions in long bones are located in the epiphysis, so curettage is first suggested to maintain joint function. If curettage is incomplete or there is a recurrence, secondary segmental resection should be considered curative. Changes of serum FGF-23 and phosphorus levels before and after the operation may be of prognostic help.
Authors: Jolanta Dadoniene; Marius Miglinas; Dalia Miltiniene; Donatas Vajauskas; Dmitrij Seinin; Petras Butenas; Tomas Kacergius Journal: World J Surg Oncol Date: 2016-01-08 Impact factor: 2.754