Malignant phosphaturic mesenchymal tumor with pulmonary metastasis

Qiu, Shui; Cao, Lili; Qiu, Yue; Yan, Pu; Li, Zi-xuan; Du, Jiang; Sun, Limei; Zhang, Qingfu

doi:10.1097/md.0000000000006750

Cited by 28 publications

(22 citation statements)

References 15 publications

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“…The clinical manifestations include hypophosphatemia and decreased serum 1,25(OH)2-vitamin D3 levels [ 8 ]. Detection of serum or tumor tissue fibroblast growth factor 23 (FGF-23) has a subsidiary value in PMT diagnosis [ 9 ]. FGF-23 is a hormone-like protein secreted by PMTs.…”

Section: Discussionmentioning

confidence: 99%

Malignant phosphaturic mesenchymal tumor-ossifying fibroma-like subtype: a case report and literature review

Qin

Zeng

et al. 2021

BMC Musculoskelet Disord

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Background A phosphaturic mesenchymal tumor (PMT) is classified into four histological subtypes: mixed connective tissue, osteoblast-like, non-ossifying fibroma-like, and ossifying fibroma-like. The ossifying fibroma-like subtype being extremely rare. Most PMTs are benign, with a minimal number becoming malignant after recurrence. In this study, we report a case of recurrence and malignant transformation of PMT-ossifying fibroma-like subtype in the left hip bone. Case presentation Here, we report the clinical manifestations, histology, pathological features, and treatment of a 57-year-old Chinese woman with a recurrent and malignant ossifying fibroma-like subtype PMT of the left iliac bone. The tumor was first discovered 3 years ago when the patient underwent surgery to remove the tumor. Precisely 2 years and 6 months after the operation, the pain in the left hip reappeared. After 6 months, the patient went to our hospital for treatment. After the tumor resection, the postoperative symptoms improved significantly, and the serum alkaline phosphatase level returned to normal. Based on clinical manifestations, evaluation of serum biochemical indicators, X-ray examination, computerized tomography scan of the pelvis, and histopathological examination of the two operations, the patient was finally diagnosed with a recurring and malignant transformation of the left iliac bone phosphaturic mesenchymal tumor-ossifying fibroma-like subtype. No tumor recurrence was found during the follow-up 15 months after the operation. Conclusions This case increases the awareness of a rare malignant subtype of PMT and provides a valuable reference for the diagnosis of this disease.

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Section: Discussionmentioning

confidence: 99%

Malignant phosphaturic mesenchymal tumor-ossifying fibroma-like subtype: a case report and literature review

Qin

Zeng

et al. 2021

BMC Musculoskelet Disord

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“…Most reported cases of PMTs are of the benign type and have either a favourable outcome or recurred locally. Cases of PMTs with distant metastases are rare and usually have evident histological features of malignancy ( 5 , 7 , 8 , 9 , 10 ) ( Table 1 ). To the best of our knowledge the current case is only the second report of PMTs with histologically benign features presenting with distant metastasis on long-term follow-up.…”

Section: Discussionmentioning

confidence: 99%

“…( 8 ) 1 Mandible No and Ki67 <3% Lung and soft tissue 24 year-course, multiple metastases, AWD Wasserman et al . ( 9 ) 2 Floor of mouth-nasal cavity (1) Yes (2) yes Lung-lung (1) >20 year-course multiple recurrences (2) >10 year-course multifocal disease Qju et al ( 7 ) 1 Right leg Yes (Ki67 >40%) Lung Recurrence after first surgery, AWNED 10 months after the last operation AWD, alive with disease; AWENED, alive with no evidence of disease; DOD, dead of disease; PMTs, phosphaturic mesenchymal tumors. …”

Section: Discussionmentioning

confidence: 99%

Distant lung metastases caused by a histologically benign phosphaturic mesenchymal tumor

Yavropoulou

Poulios

Foroulis

et al. 2018

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryTumor-induced osteomalacia (TIO) is a rare form of hypophosphatemia usually caused by phosphaturic mesenchymal tumors (PMTs); the biologic behavior of PMTs is under investigation. Herein we present a case of TIO with a protracted course over 12 years leading to a fatal outcome. A 39-year-old man presented with weakness in 2004 and was found to have decreased serum phosphorus, phosphaturia and low levels of 1,25-dihydroxyvitamin D3. Four years later he developed a painful left calf mass. The lesion was resected, but recurred causing extreme pain and dysfunction. Radiological examination showed a large cluster of soft tissue tumors affecting all the muscle compartments of the calf and a smaller lesion inside the metaphysis of the tibia. Above-knee amputation was performed. Histological examination of all lesions showed a cellular spindle cell neoplasm with variously sized vessels, wide vessel-like spaces and scattered deposits of calcified extracellular material. The tumor infiltrated skeletal muscles, subcutaneous fat and the proximal end of the fibula. The tibial lesion had identical histology. Three years after the amputation the patient presented with cough and dyspnea. Radiological examination, followed by an open biopsy, showed that there were multiple metastatic nodules of PMTs in both lungs. Shortly after the diagnosis the patient died. This case illustrates that even benign cases of PMTs may lead to a fatal outcome and the classification of PMTs into benign and malignant should be reassessed in order to correspond to its biological behavior.Learning points:PMTs, aside from having locally aggressive behavior, may metastasize and cause deathPMTs may behave aggressively despite ‘benign’ histological findings Accurate diagnosis of tumor-induced osteomalacia and patient management require a multidisciplinary approach

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“…The malignant PMTs showed features such as high cellularity, evident nuclear atypia and necrosis [ 5 , 37 ]. Recurrence and metastasis could be seen even after surgical resections and lungs as well as bones are the most frequent involved metastatic sites [ 5 , 10 , 53 ]. Distant lung metastasis has also been reported to be witnessed with a histologically benign PMT [ 53 , 54 ].…”

Section: Clinical Description and Evaluationmentioning

confidence: 99%

“…Recurrence and metastasis could be seen even after surgical resections and lungs as well as bones are the most frequent involved metastatic sites [ 5 , 10 , 53 ]. Distant lung metastasis has also been reported to be witnessed with a histologically benign PMT [ 53 , 54 ]. PMTs can present as paraneoplastic syndrome of carcinoma as well, relative reports involving ovarian cancer [ 55 ], prostate cancer [ 56 ], colon cancer [ 57 ] and lung cancer [ 58 ].…”

Section: Clinical Description and Evaluationmentioning

confidence: 99%

Tumor-induced osteomalacia

Yin

et al. 2018

Osteoporosis and Sarcopenia

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Tumor-induced osteomalacia (TIO), also known as oncogenic osteomalacia, is a rare paraneoplastic syndrome characterized by hypophosphatemia resulting from decreased tubular phosphate reabsorption, with a low or inappropriately normal level of active vitamin D. The culprit tumors of TIO could produce fibroblast growth factor 23 which plays a role in regulating renal Pi handling and 25-hydroxyvitamin D 1α-hydroxylase activity. Chronic hypophosphatemia could eventually lead to inadequate bone mineralization, presenting as osteomalacia. The diagnosis should be considered when patients manifest as hypophosphatemia and osteomalacia, or rickets and needs to be differentiated from other disorders of phosphate metabolism, such as the inhereditary diseases like X-linked hypophosphataemic rickets, autosomal dominant hypophosphataemic rickets, autosomal recessive hypophosphataemic rickets and acquired diseases like vitamin D deficiency. Localization of responsible tumors could be rather difficult since the vast majority are very small and could be everywhere in the body. A combination of thorough physical examination, laboratory tests and imaging techniques should be applied and sometimes a venous sampling may come into handy. The technology of somatostatin-receptor functional scintigraphy markedly facilitates the localization of TIO tumor. Patients undergoing complete removal of the causative neoplasm generally have favorable prognoses while a few have been reported to suffer from recurrence and metastasis. For those undetectable or unresectable cases, phosphate supplements and active vitamin D should be administrated and curative intended radiotherapy or ablation is optional.

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Malignant phosphaturic mesenchymal tumor with pulmonary metastasis

Cited by 28 publications

References 15 publications

Malignant phosphaturic mesenchymal tumor-ossifying fibroma-like subtype: a case report and literature review

Malignant phosphaturic mesenchymal tumor-ossifying fibroma-like subtype: a case report and literature review

Distant lung metastases caused by a histologically benign phosphaturic mesenchymal tumor

Tumor-induced osteomalacia

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