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首页> 外文期刊>Bone Reports >Lessons learnt from delayed diagnosis of FGF-23-producing tumour-induced osteomalacia and post-operative hungry bone syndrome
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Lessons learnt from delayed diagnosis of FGF-23-producing tumour-induced osteomalacia and post-operative hungry bone syndrome

机译:从延迟诊断到产生FGF-23产生的肿瘤诱导的骨癌和术后饥饿骨综合征的经验教训

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摘要

Tumour-induced osteomalacia (TIO) is a rare paraneoplastic syndrome caused by a fibroblast growth-factor-23 (FGF-23)-secreting phosphaturic mesenchymal tumour (PMT) and is characterised by hypophosphataemic osteomalacia. We present a 36-year-old man initially presenting with diffuse bone and joint pain who was inappropriately treated for presumed ankylosing spondylitis for 2?years. Whole-body bone scan suggested metabolic bone disease, prompting referral to our endocrine institution. He was subsequently diagnosed with persistent hypophosphataemia, inappropriately high renal tubular phosphate excretion, 1,25-dihydroxyvitamin D3suppression, severe osteoporosis and severe osteomalacia. FGF-23 concentrations (140?ng/L) were raised 3-fold above the upper limit of normal. Initial Gallium-68 (68Ga) DOTATATE positron emission tomography (PET)/CT scan missed an active lesion in the left fibular head as the field only included the mid-brain to the proximal femora. Histopathology results from tumour resection confirmed a PMT over-expressing FGF-23. Serum phosphate and FGF-23 normalised immediately post-operatively. He developed severe hypocalcaemia 3-weeks post-operatively (1.77?mmol/L) which normalised after 1?month of high-dose caltrate and calcitriol therapy. Osteomalacia, osteoporosis and associated symptoms resolved during medium-term follow-up with >100% improvement in his bone mineral density. This case report and discussion highlights the pitfalls contributing to delayed diagnosis of TIO and alerts clinicians to the potential complication of hungry bone syndrome post-tumour resection.
机译:肿瘤诱导的骨癌(TiO)是由成纤维细胞生长因子-33(FGF-23) - 分泌磷酸性间充质肿瘤(PMT)引起的罕见的肝血管塑料综合征,其特征在于脾磷血骨癌。我们展示了一名36岁的男子最初呈现弥漫性骨骼和关节疼痛,谁被认为是预测的强调脊柱炎2年的脊柱炎。全身骨骼扫描表明代谢骨病,促使我们的内分泌机构转诊。他随后被诊断出患有持续的脾磷血症,不恰当的高肾小管磷酸盐排泄,1,25-二羟基素D3抑制,严重骨质疏松症和严重的骨癌。 FGF-23浓度(140μl≤nc/ l)升高3倍,上限正常。最初的镓-68(68GA)Dotatate正电子发射断层扫描(PET)/ CT扫描错过了左侧腓骨头中的活性病变,因为该领域仅包括中大脑到近端股骨。来自肿瘤切除的组织病理学结果证实了PMT过度表达的FGF-23。血清磷酸盐和FGF-23立即标准化后可操作地。他在可操作性(1.77?mmol / l)后3周发育严重的低钙血症,其在1?月份的高剂量消融和钙硝基疗法后标准化。在中期随访期间,骨质癌,骨质疏松症和相关症状在其骨矿物密度提高的中期随访期间得到了解决。本案例报告和讨论突出了导致TiO延迟诊断的缺陷,并提醒临床医生对肿瘤后切除术后饥饿的骨综合征的潜在并发症。

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