Abstract
We describe the clinical, radiographic and histological features of skeletal involvement in four patients with end-stage renal failure due to primary oxalosis. The clinical features were unrelenting bone pain, and in two patients multiple fractures. Radiographic features were, in chronological order: (1) radiodense metaphyses and other red marrow bone; (2) cortical defects in metaphyses; (3) spontaneous fracture-separations of epiphyses of long limb bones which healed poorly. The fractures occurred through crystal deposits, and fracture displacement was associated with extrusion of crystalline material from bone. On histological examination crystals were found to replace metaphyseal bone. Pericrystalline giant cell granulomata replaced bone marrow. Erosion surfaces near granulomas were increased. Subperiosteal and intra-osseous tophi of calcium oxalate were seen. Calcium oxalate appears to precipitate with greater facility than does physiological mineral. Bone showed the features of mixed uraemic osteodystrophy in all four patients. We conclude that: (1) the fractures occurred through heavy crystal deposits; (2) ununited fractures and intra-osseous and subperiosteal tophi contributed to the pain; (3) spontaneous fractures are of poor prognostic significance. We recommend that unstable fractures be internally fixed.
Similar content being viewed by others
References
Williams HE, Smith LH Jr (1978) Primary hyperoxaluria In: Stanbury JB (ed) The metabolic basis of inherited disease. McGraw Hill, New York, pp 182–204
Hug I, Mihatch JM (1975) Die primäre Oxalose. Fortschr Röntgenstr 123:154–162
Lagier R, Revell P, Schoenboerner A (1982) Calcium oxalate deposition in growing bone: anatomical and radiological study in a case of primary oxalosis. Metab Bone Dis Rel Res 4:49–59
Benhamou CL, Pierre D, Geslin N, Viala JF, Maitre F, Chavassieux P, Edouard C, Meunier PJ (1987) Case report. Primary bone oxalosis: the roles of oxalate deposits and renal osteodystrophy. Bone 8:59–64
Gherardi G, Poggi A, Sisca S, Calderaro V, Bonucci, E (1980) Bone oxalosis and renal osteodystrophy. Arch Pathol Lab Med 104:105–111
Branaccio D, Poggi A, Cicarelli C, Bellini F, Galmozzi C, Poletti I, Maggiore Q (1981) Bone changes in end-stage oxalosis. A J R 136:935–939
Mathews M, Stauffer M, Cameron EC, Maloney N, Sherrard DJ (1979) Bone biopsy to diagnose hyperoxaluria in patients with renal failure. Ann Intern Med 90:777–779
Kalifa G, Dossans B, Gagnadoux MF, Sauvegrain J (1979) Aspects radiologiques de l'oxalose. J Radiol 60:45–49
Jahn H, Frank RM, Voegel JC, Schohn D (1980) Scanning electron microscopy and X-ray diffraction studies of human bone oxalosis. Calcif Tissue Int 30:109–119
Jacobs C, Rottembourgh J, Reachi, Le Rain M (1974) Terminal renal failure due to oxalosis in 14 patients. Proc Eur Dial Transplant Assoc 18:359–365
Julian BA, Faugere M-C, Malluche HH (1987) Case reports. Oxalosis in bone causing a radiographical mimicry of renal osteodystrophy. Am J Kidney Dis 9:436–440
Dunn HG (1955) Oxalosis. Am J Dis Child 90:58–80
Breed A, Chesney R, Friedman A, Gilbert E, Langer L, Lattoraca R (1981) Oxalosis-induced bone disease: a complication of transplantation and prolonged survival in primary hyperoxaluria. J Bone Joint Surg [Am] 63:310–316
Wiggelinkhuizen J, Fisher RM (1982) Oxalosis of bone. Pediatr Radiol 12:307–309
Melsen F, Mosekilde L (1981) The role of bone biopsy in the diagnosis of metabolic bone disease. Orthop Clin North Am 12:571–602
Schnitzler CM, Mesquita JM, Gear KA, Robson HJ, Moodley GP, Smyth AE (1990) Iliac bone biopsies at the time of periarticular stress fractures during fluoride therapy: comparison with pretreatment biopsies. J Bone Miner Res 5:141–152
Parfitt AM, Drezner MK, Glorieux FH, Kanis JA, Malluche HH, Meunier PJ, Ott SM, Recker RR (1987) Bone histomorphometry: standardisation of nomenclature, symbols, and units. J Bone Miner Res 2:595–610
Buchanan MRC, Ihle BU, Dunn CM (1981) Haemodialysis related osteomalacia: a staining method to demonstrate aluminium. J Clin Pathol 34:1352–1354
Covington TR (1984) Tetracycline. In: Covington TR, Di Palma JR, Hussar DA, Lasagna L, Tatro DS, Whitsett DL (eds) Drug facts and comparisons. Lippincott, Philadelphia, pp 1310–1321
Jowsey J (1977) Identification of bone cell activity. In: Jowsey J (ed) The bone biopsy. Plenum, New York, pp 117
Milgram JW, Salyer WR (1974) Secondary oxalosis of bone in chronic renal failure. J Bone Joint Surg [Am] 56:387–395
Ham AW (1974) Bone. In: Ham AW (ed) Histology, 7th edn. Lippincott, Philadelphia, pp 404–415
Carsen GM, Radkowski MA (1974) Calcium oxalosis. Radiology 113:165–166
Malluche HH, Faugere M-C (1986) Renal bone disease. In: Malluche HH, Faugere M-C (eds) Atlas of mineralized bone histology. Karger, Basel, pp 70–88
Scheiman JI, Najarian JS, Mauer SM (1984) Successful strategies for renal transplantation in primary oxalosis. Kidney Int 25:804–811
Watts RWE, Calnes RY, Rolles K, Danpure CJ, Morgan SH, Mansell MA, Williams R, Purkiss P (1987) Successful treatment of primary hyperoxaluria type I by combined hepatic and renal transplantation. Lancet II:474–475
Author information
Authors and Affiliations
Rights and permissions
About this article
Cite this article
Schnitzler, C.M., Kok, J.A., Jacobs, D.W.C. et al. Skeletal manifestations of primary oxalosis. Pediatr Nephrol 5, 193–199 (1991). https://doi.org/10.1007/BF01095951
Received:
Revised:
Accepted:
Issue Date:
DOI: https://doi.org/10.1007/BF01095951