Abstract
Alkaptonuria is a rare disease in which the body does not have enough of an enzyme called homogentisic acid oxidase. Osteoarthritis is the most common degenerative joint disease. Ochronotic arthritis which resulting from the deposition of oxidized homogentisic acid within the connective tissues of peripheral joints has clinical feature that resembles those of osteoarthritis, but it has a unique manifestation. We reported a case of a patient of ochronotic arthritis, arthroscopic findings showed large areas of darkly pigmented full-thickness cartilage defects in the right knee, the whole meniscal parenchymatous tissue was also darkly pigmented. Histological investigation proved to be ochronosis.
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References
Abe Y, Oshima N, Hatanaka R, Amako T, Hirohata R (1960) Thirteen cases of alkaptonuria from one family tree with special reference to osteoarthrosis alkaptonuria. J Bone Joint Surg Am 42-A:817–831
Aynaci O, Önder Ç, Turhan U (2000) Bilateral hip arthroplasty for ochronotic arthroplasty. Clin Rheumatol 19(2):150–152
Cunningham TJ, Roux E, Lagier R, Fallet GH (1989) Rapidly progressive hip osteoarthrosis. An unusual presentation of ochronosis. Clin Exp Rheumatol 7(3):315–318
de Haas V, Carbasius Weber EC, Bakker HD, Smit GP, Huijbers WA, Duran M, Poll-The BT (1998) The success of dietary protein restriction in alkaptonuria patients is age-dependent. J Inherit Metab Dis 21(8):791–798
Dieppe P, Watt I (1985) Crystal deposition in osteoarthritis: an opportunistic event? Clin Rheum Dis 11(2):367–392
Feild JR, Higley GB Sr, DeSaussure RL Jr (1963) Ochronosis with ruptured lumbar disc: case report. J Neurosurg 20:348–351
Fernandez-Canon JM, Granadino B, Beltran-Valero de Bernabe (1996) The molecular basis of alkaptonuria. Nat Genet 14(1):19–24
Forslind K, Wollheim F, Akesson B, Rydholm U (1988) Alkaptonuria and ochronosis in three siblings. Ascorbic acid treatment monitored by urinary HGA excretion. Clin Exp Rheumatol 6(3):289–292
Hamdi N, Cooke TD, Hassan B (1999) Ochronotic arthropathy: case report and review of literature. Int Orthop 23(2):122–125
Kobak AC, Oder G, Kobak S, Argin M, Inal V (2005) Ochronotic arthropathy: disappearance of alkaptonuria after liver transplantation for hepatitis B-related cirrhosis. J Clin Rheumatol 11(6):323–325
Lichtestein L, Kaplan L (1954) Hereditary ochronosis: pathologic changes observed in two necropsied cases. Am J Pathol 30(1):99–125
McCollum DE, Odom GL (1965) Alkaptonuria, ochronosis and low-back pain: a case report. J Bone Joint Surg Am 47(7):1389–1392
Millea TP, Segal LS, Liss RG, Stauffer ES (1992) Spine fracture in ochronosis: report of a case. Clin Orthop Relat Res (281):208–211
Nas K, Gür A, Akdeniz S, Cevik R, Harman M, Saraç AJ (2002) Ochronosis: a case of severe ochronotic arthropathy. Clin Rheumatol 21(2):170–172
Phornphutkul C, Introne WJ, Perry MB, Bernardini I, Murphey MD, Fitzpatrick DL, Anderson PD, Huizing M, Anikster Y, Gerber LH, Gahl WA (2002) Natural history of alkaptonuria. N Engl J Med 347(26):2111–2121
Touart DM, Sau P (1998) Cutaneous deposition diseases. J Am Acad Dermatol 39(4 Pt 1):527–544
Wolff JA, Barshop B, Nyhan WL, Leslie J, Seegmiller JE, Gruber H, Garst M, Winter S, Michals K, Matalon R (1989) Effects of ascorbic acid in alkapronuria: alterations in benzoquinone acetic acid and an ontogenic effect in infancy. Pediatr Res 26(2):140–144
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Zhao, B.H., Chen, B.C., Shao, D.C. et al. Osteoarthritis? Ochronotic arthritis!. Knee Surg Sports Traumatol Arthrosc 17, 778–781 (2009). https://doi.org/10.1007/s00167-009-0778-0
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DOI: https://doi.org/10.1007/s00167-009-0778-0