Tumor-induced osteomalacia caused by head and neck tumors: report of an unusual case and literature review
Tumor-induced osteomalacia caused by head and neck tumors: report of an unusual case and literature review
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头颈部肿瘤引起的肿瘤性骨软化症一例罕见病例报告及文献复习
DOI：
10.21037/fomm-21-23
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发表时间：
2021-06
期刊：
影响因子：
--
通讯作者：
Wang L
中科院分区：
文献类型：
--
作者：
Shi JC;Wang LY;Abdelrehem A;Zhang Y;Wu ZQ;Tian Z;Li SY;Wang L
肿瘤引起的骨软化症是一种相对罕见的副肿瘤综合征。全世界仅有不到200例头颈部肿瘤后肿瘤引起的骨软化症的报道。在此，我们报告一例由颌面部肿瘤引起的肿瘤相关性骨软化，并复习相关文献，以提高临床医生对肿瘤所致骨软化的认识，并提出诊断策略。在此病例中，患者首先出现左侧大腿区域的骨痛，后来累及肋骨和脚踝，被错误地视为强直性脊柱炎，没有任何明显的效果。后来症状恶化，患者无法行走。实验室检查显示，血清磷降低，碱性磷酸酶和甲状旁腺激素升高。此外，25羟基维生素D和血钙正常。患者在另一家医院被诊断为骨软化症，随后的正电子发射计算机断层扫描显示左侧上颌骨局限性骨质破坏，放射性摄取增加。入院后行“左上颌骨肿块切除+左上颌窦根治术”。术后病理表现为磷尿性间充质瘤，上皮型和结缔组织混合型。术后第4天，患者血磷水平恢复正常，骨痛感和关节僵硬程度逐渐改善，患者较术前柔韧性增强。外科医生从这一病例报告中可以学到以下几点：(I)如果发现骨软化症，应仔细区分是遗传性的还是继发性的，仔细询问是否有家族史和特殊病史，并仔细考虑体检和影像评估，尽快定位诱发肿瘤；(Ii)肿瘤性骨软化症多为良性或低度恶性，高度恶性的发生率较低。然而，由于其局部侵袭性，经常在肿瘤周围进行广泛的切除以减少复发。总之，这份病例报告是值得的，因为它解决了这种病例的常见误导性陈述以及随后被误认为骨质疏松症的情况，这反过来会影响预后。
Tumor-induced osteomalacia is a relatively rare paraneoplastic syndrome. Fewer than 200 cases of tumor-induced osteomalacia following head and neck tumors have been reported worldwide. Herein, we report a case of tumor-related osteomalacia caused by a maxillofacial tumor, and review the literature in order to enhance clinicians’ understanding of tumor-induced osteomalacia and propose diagnostic strategies. In this case, the patient presented with bone pain first in the left thigh region and later involved the ribs and ankle, which had been mistakenly treated as being ankylosing spondylitis without any obvious effect. The symptoms worsened later and the patient was unable to walk. Laboratory tests showed that serum phosphorus decreased, while alkaline phosphatase and parathyroid hormone increased. Moreover, 25 hydroxyvitamin D and serum calcium were normal. The patient was diagnosed as osteomalacia in another hospital, and the subsequent positron emission tomography computed tomography examination suggested focal bony destruction of the left maxilla with an increased radioactive uptake. After hospital admission, the patient underwent “resection of the left maxillary mass + radical resection of the left maxillary sinus”. The postoperative pathology indicated phosphaturic mesenchymal tumor; mixed epithelial and connective tissue type. On the 4th postoperative day, the serum phosphorus level of the patient returned to normal, the sensation of bone pain and joint stiffness gradually improved, and the patient became more flexible than before surgery. Surgeons can learn the followings from this case report: (I) if osteomalacia is found, we should carefully distinguish whether it is genetic or secondary, carefully inquire whether there is a family history and special medical history, and carefully consider physical examination and imaging evaluation to localize the inducing tumor as soon as possible; (II) tumor-induced osteomalacia is mostly benign or lowgrade malignancy, with a rare incidence of high-grade malignancy. However, due to its local invasive nature, an extensive resection is often performed around the tumor to reduce recurrence. In a word, this case report is worthy that it addresses the common misleading presentation of such cases and subsequent mistreatment as osteoporosis that inversely affects prognosis.
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DOI：
10.3760/j.issn:0529-5807.2005.11.006
发表时间：
2005-11
期刊：
Zhonghua bing li xue za zhi = Chinese journal of pathology
影响因子：
--
作者：
D. Zhong;Tong-hua Liu;Di Yang;R. Feng;Q. Cui;Yufeng Luo;Yong Jia
通讯作者：
D. Zhong;Tong-hua Liu;Di Yang;R. Feng;Q. Cui;Yufeng Luo;Yong Jia
影响因子：
2.1
作者：
H. Qari;Aya Hamao-Sakamoto;Clay Fuselier;Y. Cheng;H. Kessler;John M. Wright
通讯作者：
H. Qari;Aya Hamao-Sakamoto;Clay Fuselier;Y. Cheng;H. Kessler;John M. Wright
DOI：
--
发表时间：
1964
期刊：
Gan no rinsho. Japan journal of cancer clinics
影响因子：
--
作者：
I. Aoike
通讯作者：
I. Aoike
影响因子：
3.3
作者：
Ito, Nobuaki;Fukumoto, Seiji;Fujita, Toshiro
通讯作者：
Fujita, Toshiro
影响因子：
--
作者：
Lewiecki, E. Michael;Urig, Edward J., Jr.;Williams, Ralph C., Jr.
通讯作者：
Williams, Ralph C., Jr.