Total Knee Replacement in Alkaptonuric Ochronosis
Two Case Reports and Literature Review
Keywords:
alkaptonuria, ochronotic arthropathy, total knee replacementAbstract
Background and objective: Alkaptonuria is a rare disease characterized by the accumulation of homogentisic acid (HGA). Over time, these patients may develop disabling ochronotic arthropathy. We present 2 cases of patients with end-stage arthropathy treated with total knee arthroplasty (TKA).
Methods: Both patients complained of disabling knee pain and reported limited walking distance (200-300 m). One had a history of osteotomy for medial knee arthtritis and ignored his underlying condition. The other presented with valgus gonoarthrosis and diagnosis of alkaptonuria.
Results: Intraoperatively, the characteristic dark-blue color in the joint was observed. Both patients evolved favorably after TKA with excellent results according to the Knee Society Scores (KSS) at three years of follow-up.
Conclusion: We believe TKA is the right treatment for patients with end-stage disease because it offers considerable relief from pain and allows patients to recover function.
References
Karaoglu S., Karaaslan F., Mermerkaya MU. Long-term result of arthroplasty in the treatment of a case of ochronotic arthropathy. Acta Orthop Traumatol Turc 2016;50:584–6.
Millucci L., Giorgetti G., Viti C., Ghezzi L., Gambassi S., Braconi D. Chondroptosis in alkaptonuric cartilage. J Cell Physiol 2015;230:1148–57.
Millucci L., Bernardini G., Spreafico A., Orlandini M., Braconi D., Laschi M. Histological and ultrastructural characterization of alkaptonuric tissues. Calcif Tissue Int 2017;101:50–6.
Li N., Tian W., Yuan Q., He D. Cervical spondylotic myelopathy due to the ochronotic arthropathy of the cervical spine. J Korean Neurosurg Soc 2016;59(1):65-68.
Gil J.A., Wawrzynski J., Waryasz GR. Orthopedic manifestations of ochronosis: pathophysiology, presentation, diagnosis, and management. Am J Med 2016;129(5):536.e1–6.
Ewald FC. The knee socity total knee arthroplasty roentgenographic evaluation and scoring system. Clin Orthop Relat Res 1989;248:9-12.
Harun M., Hayrettin Y., Serhat M., Cuneyt M., Fırat F., Ufuk O. A rare cause of arthropathy: an ochronotic patient with black joints. Int J Surg Case Rep 2014;5:554–7.
Drakoulakis E., Varvitsiotis D., Psarea G., Feroussis J. Ochronotic arthropathy: diagnosis and management—a critical review. Am J Orthop 2012;41:80–3.
Keller J.M., Macaulay W., Nercessian O.A., Jaffe I.A. New developments in ochronosis: review of the literature. Rheumatol Int. 2005;25:81–85.
Raaijmaakers M., Steenbrugge F., Dierickx C. Ochronosis, arthroscopy of a black knee: a case report and review of the literature. Knee Surg Sports Traumatol Arthrosc. 2008;16:182–184.
Laskar F.H., Sargison K.D. Ochronotic arthropathy. A review with four case reports. J Bone Jt Surg. 1970;52:653–666.
Damarla N., Linga P., Goyal M., Tadisina SR., Reddy GS, Bommisetti H. Alkaptonuria: a case report. Indian J Ophthalmol 2017;65:518–21.
Acar M.A., Erkocak O.F., Aydin B.K., Altan E., Senaran H., Elmadag N.M. Pacientes con cadera negra y rodilla negra debido a artropatía ocrónica: reporte de caso y revisión de la literatura. Oman Med J. 2013; 28(6): 448–9.
Couto A., Rodrigues A, Oliveira P., and Seara M. Ochronotic arthropathy - a rare clinical case. Oxf Med Case Reports 2018;9:302-305.
Spencer J.M., Gibbons C.L., Sharp R.J., Carr A.J., Athanasou N.A. Arthroplasty for ochronotic arthritis: no failure of 11 replacements in three patients followed for 6–12 years. Acta Orthop Scand. 2004;75:355–358.
Wu Chean Lee, Tong Leng Tan, Ying Ho Chan. Total Knee Arthroplasty in Ochronosis Arthropathy: A case Report and Systematic Review. Case Reports Orthopedics. 2019;2019:1-8.
Rodriguez J. A., Saddler S., Edelman S., Ranawar C. S. Long-term results of total knee arthroplasty in class 3 and 4 rheumatoid arthritis. The Journal of Arthroplasty. 1996;11(2):141-145.
Ulucay C, Ozler T, Altintas F, Inan M, Onur A, et al. Arthroplasty in ochronosis “tips and pearls in surgery”: case series. J Arthritis. 2013;3;1-3.
Kajino A., Yoshino S., Kohda M., Nagashima S. Comparision of the results of bilateral total knee arthroplasty with and without patellar replacement for rheumatoid arthritis. A follow-up note. The Journal of Bone and Joint Surgery. American Volume. 1997;79(4):570-574.
Downloads
Published
Issue
Section
License
Copyright (c) 2021 Publisher

This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Transfer of Copyright and Permission to Reproduce Parts of Published Papers.
Authors retain the copyright for their published work. No formal permission will be required to reproduce parts (tables or illustrations) of published papers, provided the source is quoted appropriately and reproduction has no commercial intent. Reproductions with commercial intent will require written permission and payment of royalties.