1. Prolonged Hypophosphatemia and Intensive Care After Curative Surgery of Tumor Induced Osteomalacia: A Case Report
- Author
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Camilla Schalin-Jäntti, Eeva Ryhänen, Niina Matikainen, HUS Abdominal Center, Endokrinologian yksikkö, University of Helsinki, Helsinki University Hospital Area, and Clinicum
- Subjects
Fibroblast growth factor 23 ,oncogenic osteomalacia ,medicine.medical_specialty ,Endocrinology, Diabetes and Metabolism ,FRACTURE RISK ,Case Report ,030209 endocrinology & metabolism ,Physical examination ,tumor-induced hypophosphatemia ,THERAPY ,Diseases of the endocrine glands. Clinical endocrinology ,fibroblast growth factor 23 ,03 medical and health sciences ,Endocrinology ,0302 clinical medicine ,Intensive care ,medicine ,FAILURE ,intensive care ,030304 developmental biology ,0303 health sciences ,Osteomalacia ,medicine.diagnostic_test ,perioperative hypophosphatemia ,tumor-induced osteomalacia ,business.industry ,WOMEN ,Muscle weakness ,Perioperative ,RC648-665 ,surgical complications ,medicine.disease ,3. Good health ,Oncogenic osteomalacia ,Surgery ,ROSIGLITAZONE ,3121 General medicine, internal medicine and other clinical medicine ,medicine.symptom ,BONE ,business ,Hypophosphatemia - Abstract
IntroductionRare FGF23-producing mesenchymal tumors lead to paraneoplastic tumor-induced osteomalacia (TIO) presenting with phosphate wasting, hypophosphatemia, chronic hypomineralization of the bone, fragility fractures and muscle weakness. Diagnosis of TIO requires exclusion of other etiologies and careful search for a mesenchymal tumor that often is very small and can appear anywhere in the body. Surgical removal of the tumor is the only definitive treatment of TIO. Surgical complications due to chronic hypophosphatemia are not well recognized.Case DescriptionThe current case describes severe fragility fractures in a 58-year-old woman, who lost her ability to walk and was bedridden for two years. First, the initial diagnostic laboratory work-up did not include serum phosphorus measurements, second, the suspicion of adverse effects of pioglitazone as an underlying cause delayed correct diagnosis for at least two years. After biochemical discovery of hyperphosphaturic hypophosphatemia at a tertiary referral centre, a FGF23-producing tumor of the mandible was discovered on physical examination, and then surgically removed. Postoperatively, severe hypophosphatemia and muscle weakness prolonged the need for ventilation support, intensive care and phosphate supplementation. After two years of rehabilitation, the patient was able to walk short distances. The tumor has not recurred, and serum phosphate concentration has remained within normal limits during 3.5 years of follow-up.ConclusionsThe case report illustrates knowledge gaps in the diagnostic work-up of rare causes of low bone mass and fragility fractures. Compared to other low phosphate conditions, surgical recovery from TIO-induced hypophosphatemia warrants special attention. Increased alkaline phosphatase concentration may indicate impaired postsurgical recovery due to prolonged hypophosphatemia, underlining the need for proactive perioperative correction of hypophosphatemia.
- Published
- 2021
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