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.
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Abbreviations
- ENS :
-
epidermal naevus syndrome
- TRP :
-
tubular phosphate reabsorption
- VDRR:
-
vitamin D resistant hypophosphataemic rickets
References
Amir G, Boneh A, Tochner Z (1993) Widespread hemangiomatosis of bone associated with rickets: Recovery after irradiation. J Pediatr 123: 269–272
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: 56–60
Asnes RS, Berdon WE, Basset CA (1981) Hypophosphatemic rickets in an adolescent cured by excision of a nonossifying fibroma. Clin Pediatr 20: 646–648
Balsan S, Guivarch J, Dartois A-M (1967) Rachitisme vitamino-resistant associé à une neurofibromatose probable chez un enfant. Arch Franç Pediatr 24: 609–632
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: 1645–1649
Carey DE, Drezner MK, Hamdam JA (1986) Hypophosphatemic rickets/osteomalacia in linear sebaceous nevus syndrome: a variant of tumor-induced osteomalacia. J Pediatr 109: 994–1000
Dent CE, Gertner EM (1976) Hypophosphatemic osteomalacia in fibrous dysplasia. OJ Med 69: 411–420
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: 873–876
Hanukoglu A, Chalew SA, Sun CJ (1989) Surgically curable hypophosphatemic rickets. Clin Pediatr 28: 321–325
Harrison HE (1973) Oncogenous rickets: Possible elaboration by a tumor of a humoral substance inhibiting tubular reabsorption of phosphate. Pediatrics 52: 432–433
Konishi K, Nakamura M, Yamakawa H (1991) Case report: hypophosphatemic osteomalacia in von Recklinghausen neurofibromatosis. Am J Med Sci 301: 322–328
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: 1205–1206
Moncrieff MW, Brenton DP, Arthur LJH (1978) Case of tumor rickets. Arch Dis Child 53: 740–745
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: 46–53
Nuovo MA, Dorfman HD, Sun CJ (1989) Tumor-induced osteomalacia and rickets. Am J Surg Pathol 13(7): 588–599
Parker MS, Klein I, Haussler MR (1981) Tumor-induced osteomalacia: evidence of a surgically correctable alteration in vitamin D metabolism. JAMA 245: 492–493
Pollack JA, Schiller AL, Crawford JD (1973) Rickets and myopathy cured by removal of a nonossifying fibroma of bone. Pediatrics 52: 364–371
Popovtzer MM (1981) Tumor-induced hypophosphatemic osteomalacia: evidence for a phosphaturic cyclic AMP-independent action of tumor extract (abstract). Clin Res 29: 418A
Prader A, Illig R, Uehlinger RE (1959) Rachitis infolge Knochentumors. Acta Helvetica Pediatrica 14: 554–565
Scriver CR, Tenehouse HS, Glorieux FH (1991) X-linked hypophosphatemia: An appreciation and a survey of progress since 1958. Classics in Medicine 218–228
Stanbury SW (1972) Tumor-associated hypophosphatemic oseomalacia and rickets. Clin Endocrinol (Oxf) 1: 256–266
Weidner N (1991) Review and update: oncogenic osteomalacia-rickets. Ultrastruct Pathol 15: 317–333
Weidner N, Bar RS, Weiss D (1985) Neoplastic pathology of oncogenic osteomalacia/rickets. Cancer 55: 1691–1705
Weidner N, Santa Cruz D (1987) Phosphaturic mesenchymal tumors. A polymorphous group causing osteomalacia or rickets. Cancer 59: 1442–1454
Wilhoite DR (1975) Acquired rickets and solitary bone tumor: the question of a causal relationship. Clin Orthop 109: 210–211
Yoshikawa S, Nakamura T, Takagi M (1977) Benign osteoblastoma as a cause of osteomalacia. A report of two cases. J Bone Joint Surg 59B: 279–286
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Eyskens, B., Proesmans, W., Van Damme, B. et al. Tumour-induced rickets: A case report and review of the literature. Eur J Pediatr 154, 462–468 (1995). https://doi.org/10.1007/BF02029356
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DOI: https://doi.org/10.1007/BF02029356