Day Alvin Lee, Gutiérrez Orlando M, Guthrie Barton L, Saag Kenneth G
Division of Clinical Immunology and Rheumatology, Department of Medicine, University of Alabama at Birmingham, 1720 2nd Avenue South, FOT 839, 35294 Birmingham, AL, USA.
Division of Nephrology, Department of Medicine, University of Alabama at Birmingham, 35294 Birmingham, AL, USA.
Joint Bone Spine. 2020 Jan;87(1):81-83. doi: 10.1016/j.jbspin.2019.07.012. Epub 2019 Aug 3.
Tumor-induced osteomalacia is a rare cause of acquired hypophosphatemia due to the paraneoplastic overproduction of fibroblast growth factor-23. Unlike many causes of osteomalacia, tumor-induced osteomalacia is curable by resection of the offending tumor. If a patient has a tumor that is unidentifiable, unresectable, or makes the decision to forgo surgery, medical treatment is recommended. Burosumab (KRN23) is a fully human monoclonal antibody against fibroblast growth factor-23 that was recently approved for the treatment of X-linked hypophosphatemia. We present a case of tumor-induced osteomalacia due to two somatostatin receptor avid meningiomas. The patient initially was wheelchair bound due to symptoms of diffuse bone and muscle pain with recurrent traumatic and nontraumatic fractures. Serum phosphate was 1.8mg/dL (reference range: 2.4-5.0mg/dL) with no other laboratory or historical cause. Workup revealed two widely separated intracranial meningiomas with typical magnetic resonance imaging characteristics. The duplicity of tumors precluded safe surgery and the potential delay in, or lack of, efficacy using radiosurgery prompted the treatment team to opt for medical treatment. Burosumab was initiated resulting in improvement in pain symptoms and mobility. Serum phosphate normalized. Trials are ongoing to assess the utility of burosumab in the treatment of tumor-induced osteomalacia.
肿瘤诱导的骨软化症是一种罕见的获得性低磷血症病因,由成纤维细胞生长因子-23的副肿瘤性过度产生所致。与许多骨软化症病因不同,肿瘤诱导的骨软化症可通过切除致病肿瘤治愈。如果患者有无法识别、无法切除的肿瘤,或决定放弃手术,则建议进行药物治疗。布罗索尤单抗(KRN23)是一种针对成纤维细胞生长因子-23的全人单克隆抗体,最近被批准用于治疗X连锁低磷血症。我们报告一例由两个生长抑素受体亲和性脑膜瘤引起的肿瘤诱导的骨软化症病例。患者最初因弥漫性骨和肌肉疼痛症状以及反复出现的创伤性和非创伤性骨折而需依靠轮椅行动。血清磷酸盐为1.8mg/dL(参考范围:2.4 - 5.0mg/dL),无其他实验室检查或病史原因。检查发现两个相距甚远的颅内脑膜瘤,具有典型的磁共振成像特征。肿瘤的双重性使得安全手术无法进行,而使用放射外科手术可能延迟或缺乏疗效促使治疗团队选择药物治疗。开始使用布罗索尤单抗后,疼痛症状和活动能力得到改善。血清磷酸盐恢复正常。目前正在进行试验以评估布罗索尤单抗在治疗肿瘤诱导的骨软化症中的效用。