Treatment and clinical features of tumor-induced osteomalacia:a retrospective review of fifteen patients

LI Wei; WANG Xiangdang; ZUO Qingyao; WANG Hong; HUANG Yanhong; SONG Hui; and DENG Wei.

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Medical Journal of the Chinese People Armed Police Forces ›› 2018, Vol. 29 ›› Issue (6) : 566-570.

ORIGINAL ARTICLES

Treatment and clinical features of tumor-induced osteomalacia:a retrospective review of fifteen patients

LI Wei¹,WANG Xiangdang²,ZUO Qingyao¹,WANG Hong¹,HUANG Yanhong³,SONG Hui³,and DENG Wei¹.

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Abstract

Objective To investigate the clinical features, diagnostic process and outcomes of tumor-induced osteomalacia (TIO).Methods A retrospective analysis of fifteen patients with TIO was performed, involving pre- and post-operative clinical features and biochemical changes.Results Among the fifteen patients, there were two cases of recurrence (13.3%)and one case of metastases (6%)after recurrence. The average age was (42.8±10.7 )years, and the mean duration of disease was (3.5±2.8 )years. Serum phosphorus concentrations were low in all these patients (0.41±0.10)mmol/L.The renal phosphate threshold (RTPC) decreased in all the patients with an average of (0.33±0.10) mmol/L. These patients were treated surgically nineteen times. Nine tumors were found in soft tissues and ten tumors in bones. Thirteen of these cases (86.7%) were cured through primary operation and serum phosphorus concentrations returned to normal in (3.7±1.6 )days. All the nineteen tumors were phosphaturic mesenchymal tumors of the mixed connective tissue variant. One case was found to be malignant during the follow-up. Compared with tumors in bones, tumors in soft tissues caused a lower serum phosphorus concentration and lower RTPC (t=2.264, P=0.041). Regression analysis showed that the blood phosphorus concentration was influenced primarily by RTPC (standardized beta coefficient=0.836, t=3.862,P=0.005), and that RTPC was influenced by tumor volume and tumor tissue origin (standardized beta coefficient=0.405、0.546,t=-2.635、3.925,P=0.030、0.004, respectively).Conclusions The chance of recurrence and metastasis of TIO is high. Compared with 24-hour urine phosphorus concentrations, RTPC is more suitable for the evaluation of impaired renal tubular function. Patients with a tumor in soft tissues are in more critical condition.

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tumor-induced osteomalacia / malignancy / recurrence / hypophosphatemia / phosphaturic mesenchymal tumor

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LI Wei,WANG Xiangdang,ZUO Qingyao,WANG Hong,HUANG Yanhong,SONG Hui,and DENG Wei.. Treatment and clinical features of tumor-induced osteomalacia:a retrospective review of fifteen patients[J]. Medical Journal of the Chinese People Armed Police Forces. 2018, 29(6): 566-570

References

[1] Kumar R,Folpe A L, Mullan B P. Tumor-induced osteomalacia[J].Transl Endocrinol Metab,2015,7(3):1-23.
[2] Berndt T J, Schiavi S, Kumar R."Phosphatonins" and the regulation of phosphorus homeostasis[J].Am J Physiol Renal Physiol,2005,289(6):F1170-1182.
[3] Hautmann A H, Hautmann M G, Klbl O, et al.Tumor-Induced Osteomalacia: an up-to-date review[J].Curr Rheumatol Rep, 2015,17(6):512.
[4] Euhus D, Hudd C, LaRegina M. Tumor measurement in the nude mouse[J].J Surg Oncol,1986,31: 229-234.
[5] Walton R J, Bijvoet O L. Nomogram for derivation of renal thresholdphosphate concentration[J]. Lancet,1975,2(7929):309-310.
[6] Florenzano P, Gafni R I, Collins M T. Tumor-induced osteomalacia[J].Bone Rep,2017,209(7):90-97.
[7] Minisola S, Peacock M, Fukumoto S,et al.Tumor-induced osteomalacia[J].Nat Rev Dis Primers,2017,3(17044):1-15.
[8] Feng J,Jiang Y,Wang O,et al.The diagnostic dilemma of tumor induced osteomalacia:a retrospective analysis of 144 cases[J].Endocr J,2017, 64(7):675-683.
[9] Gore M O, Welch B J, Geng W,et al.Renal phosphate wasting due to tumor-induced osteomalacia: a frequently delayed diagnosis[J].Kidney Int, 2009,76(3):342-347.
[10] Sun Z J,Jin J,Qiu G X,et al.Surgical treatment of tumor-induced osteomalacia: a retrospective review of 40 cases with extremity tumors[J].BMC Musculoskelet Disord,2015,16(43):1-8.
[11] Breer S, Brunkhorst T, Beil F T,et al.68Ga DOTA-TATE PET/CT allows tumor localization in patients with tumor-induced osteomalacia but negative 111In-octreotide SPECT/CT[J].Bone,2014,64:222-227.
[12] Singh D, Chopra A, Ravina M,et al.Correction to oncogenic osteomalacia: role of Ga-68 DOTANOC PET/CT scan in identifying the culprit lesion and its management[J].Br J Radiol,2017,90(1079):20160811e.
[13] Lee J Y, Park H S, Han S,et al.Localization of oncogenic osteomalacia by systemic venous sampling of fibroblast growth factor 23[J].Yonsei Med J,2017,58(5):981-987.
[14] Bhadada S K,Palnitkar S,Qiu S,et al.Deliberate total parathyroidectomy: a potentially novel therapy for tumor-induced hypophosphatemic osteomalacia[J].J Clin Endocrinol Metab,2013,98(11):4273-4278.
[15] Seufert J, Ebert K, Müller J, et al.Octreotide therapy for tumor-induced osteomalacia[J].N Engl J Med,2001,345(26):1883-1888.
[16] Tella S H, Amalou H, Wood B J,et al.Multimodality image-guided cryoablation for inoperable tumor-induced osteomalacia[J].J Bone Miner Res,2017,32(11):2248-2256.
[17] Honda R, Kawabata Y, Ito S.Phosphaturic mesenchymal tumor, mixed connective tissue type, non-phosphaturic variant: report of a case and review of 32 cases from the Japanese published work[J].J Dermatol,2014,41(9):845-849.
[18] Lee J C, Su S Y, Changou C A,et al.Characterization of FN1-FGFR1 and novel FN1-FGF1 fusion genes in a large series of phosphaturic mesenchymal tumors[J].Mod Pathol,2016,29(11):1335-1346.
[19] Zhang Q, Doucet M, Tomlinson R E,et al.The hypoxia-inducible factor-1α activates ectopic production of fibroblast growth factor 23 in tumor-induced osteomalacia[J].Bone Res,2016,4:16011.