Tumour-induced rickets: A case report and review of the literature
Hematology/Oncology Original Paper
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Abstract
Hypophosphataemic rickets was diagnosed in a 6-year-old boy with a negative family history. After 16 years of medical treatment he developed a malignant sarcoma of the right distal thigh. Removal of the tumour by high amputation of the leg resulted in disappearance of the phosphate leak. In spite of surgery and chemotherapy, the patient died due to extensive lung metastases. Retrospective analysis of the initial X-ray films showed a benign lesion on the lateral side of the right distal femur. This lesion is believed to be at the origin of the rickets. This is the first paediatric case reported with malignant degeneration of a benign tumour causing rickets.
Conclusion
Patients with the classical hallmarks of X-linked, familial hypophosphataemic rickets but no affected family members should have a careful periodic search for a tumour, even years after onset of the disorder.
Key words
Tumour-induced rickets Hypophosphataemic rickets Phosphaturic mesenchymal tumours Phosphaturic factorAbbreviations
- ENS
epidermal naevus syndrome
- TRP
tubular phosphate reabsorption
- VDRR
vitamin D resistant hypophosphataemic rickets
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References
- 1.Amir G, Boneh A, Tochner Z (1993) Widespread hemangiomatosis of bone associated with rickets: Recovery after irradiation. J Pediatr 123: 269–272PubMedGoogle Scholar
- 2.Aschinberg LC, Solomon LM, Zeis PM (1977) Vitamin D-resistant rickets associated with epidermal nevus syndrome: demonstration of a phosphaturic substance in the dermal lesions. J Pediatr 91: 56–60PubMedGoogle Scholar
- 3.Asnes RS, Berdon WE, Basset CA (1981) Hypophosphatemic rickets in an adolescent cured by excision of a nonossifying fibroma. Clin Pediatr 20: 646–648Google Scholar
- 4.Balsan S, Guivarch J, Dartois A-M (1967) Rachitisme vitamino-resistant associé à une neurofibromatose probable chez un enfant. Arch Franç Pediatr 24: 609–632Google Scholar
- 5.Cai Q, Hodgson SF, Kao PC (1994) Brief report. Inhibition of renal phosphate transport by a tumor product in a patient with oncogenic osteomalacia. N Engl J Med 330: 1645–1649PubMedGoogle Scholar
- 6.Carey DE, Drezner MK, Hamdam JA (1986) Hypophosphatemic rickets/osteomalacia in linear sebaceous nevus syndrome: a variant of tumor-induced osteomalacia. J Pediatr 109: 994–1000PubMedGoogle Scholar
- 7.Dent CE, Gertner EM (1976) Hypophosphatemic osteomalacia in fibrous dysplasia. OJ Med 69: 411–420Google Scholar
- 8.Fukumoto Y, Tarui S, Tsukiyama K (1979) Tumor-induced Vitamin D-resistant hypophosphatemic osteomalacia associated with proximal renal tubular dysfunction and 1.25-Dihydroxyvitamin deficiency. J Clin Endocrinol Metab 49: 873–876PubMedGoogle Scholar
- 9.Hanukoglu A, Chalew SA, Sun CJ (1989) Surgically curable hypophosphatemic rickets. Clin Pediatr 28: 321–325Google Scholar
- 10.Harrison HE (1973) Oncogenous rickets: Possible elaboration by a tumor of a humoral substance inhibiting tubular reabsorption of phosphate. Pediatrics 52: 432–433PubMedGoogle Scholar
- 11.Konishi K, Nakamura M, Yamakawa H (1991) Case report: hypophosphatemic osteomalacia in von Recklinghausen neurofibromatosis. Am J Med Sci 301: 322–328PubMedGoogle Scholar
- 12.Martini A, Notarangelo LD, Barberis L (1983) Acquired vitamin D-resistant rickets caused by prolonged latency in appearance of bone tumor. Am J Dis Child 137: 1205–1206PubMedGoogle Scholar
- 13.Moncrieff MW, Brenton DP, Arthur LJH (1978) Case of tumor rickets. Arch Dis Child 53: 740–745PubMedGoogle Scholar
- 14.Miyauchi A, Fukase M, Tsutsumi M (1988) Hemangiopericytoma induced osteomalacia: tumor transplantation in nude mice causes hypophosphatemia and tumor extracts inhibit renal 25-hydroxyvitamin D 1-hydroxylase activity. J Clin Endocrinol Metab 67: 46–53PubMedGoogle Scholar
- 15.Nuovo MA, Dorfman HD, Sun CJ (1989) Tumor-induced osteomalacia and rickets. Am J Surg Pathol 13(7): 588–599PubMedGoogle Scholar
- 16.Parker MS, Klein I, Haussler MR (1981) Tumor-induced osteomalacia: evidence of a surgically correctable alteration in vitamin D metabolism. JAMA 245: 492–493CrossRefPubMedGoogle Scholar
- 17.Pollack JA, Schiller AL, Crawford JD (1973) Rickets and myopathy cured by removal of a nonossifying fibroma of bone. Pediatrics 52: 364–371PubMedGoogle Scholar
- 18.Popovtzer MM (1981) Tumor-induced hypophosphatemic osteomalacia: evidence for a phosphaturic cyclic AMP-independent action of tumor extract (abstract). Clin Res 29: 418AGoogle Scholar
- 19.Prader A, Illig R, Uehlinger RE (1959) Rachitis infolge Knochentumors. Acta Helvetica Pediatrica 14: 554–565Google Scholar
- 20.Scriver CR, Tenehouse HS, Glorieux FH (1991) X-linked hypophosphatemia: An appreciation and a survey of progress since 1958. Classics in Medicine 218–228Google Scholar
- 21.Stanbury SW (1972) Tumor-associated hypophosphatemic oseomalacia and rickets. Clin Endocrinol (Oxf) 1: 256–266Google Scholar
- 22.Weidner N (1991) Review and update: oncogenic osteomalacia-rickets. Ultrastruct Pathol 15: 317–333PubMedGoogle Scholar
- 23.Weidner N, Bar RS, Weiss D (1985) Neoplastic pathology of oncogenic osteomalacia/rickets. Cancer 55: 1691–1705PubMedGoogle Scholar
- 24.Weidner N, Santa Cruz D (1987) Phosphaturic mesenchymal tumors. A polymorphous group causing osteomalacia or rickets. Cancer 59: 1442–1454PubMedGoogle Scholar
- 25.Wilhoite DR (1975) Acquired rickets and solitary bone tumor: the question of a causal relationship. Clin Orthop 109: 210–211Google Scholar
- 26.Yoshikawa S, Nakamura T, Takagi M (1977) Benign osteoblastoma as a cause of osteomalacia. A report of two cases. J Bone Joint Surg 59B: 279–286Google Scholar
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