Black Femoral Head Due to Ochronotic Arthropathy: A Case Report
Abstract
Background: Ochronotic arthropathy is a rare complication in patients with alkaptonuria (AKU) that occurs as a result of ochronotic pigment accumulation in the joints.
Case Report: This case report presents a 54-year-old patient with severe degenerative arthritis who underwent total hip arthroplasty on both sides within an interval of three years. The diagnosis was made by observation of a dark capsule and femoral head during the second surgery. The surgical treatments significantly reduced the pain and improved the range of motion (ROM).
Conclusion: Orthopedic surgeons must be vigilant about clinical manifestations of this rare condition, before and during the surgery. Arthroplasty is a favourable therapeutic option in patients suffering from ochronotic arthropathy.
1. Jiang L, Cao L, Fang J, Yu X, Dai X, Miao X. Ochronotic arthritis and ochronotic Achilles tendon rupture in alkaptonuria: A 6 years follow-up case report in China. Medicine (Baltimore). 2019;98(34):e16837. doi: 10.1097/MD.0000000000016837. [PubMed: 31441856]. [PubMed Central: PMC6716692].
2. Schiavone ML, Pecorelli A, Woodby B, Ferrara F, Pambianchi E, Santucci A, et al. Mechanisms involved in the unbalanced redox homeostasis in osteoblastic cellular model of Alkaptonuria.
Arch Biochem Biophys. 2020;690:108416. doi:
10.1016/j.abb.2020.108416. [PubMed: 32502471].
3. Helliwell TR, Gallagher JA, Ranganath L. Alkaptonuria-a review of surgical and autopsy pathology. Histopathology.
2008;53(5):503-12.doi:10.1111/j.1365-2559.2008.03000.x.
[PubMed: 18336562].
4. Martin JP Jr, Batkoff B. Homogentisic acid autoxidation and oxygen radical generation: Implications for the etiology of alkaptonuric arthritis. Free Radic Biol Med. 1987;3(4):241-50. doi: 10.1016/s0891-5849(87)80031-x. [PubMed: 3121448].
5. Taylor AM, Boyde A, Wilson PJ, Jarvis JC, Davidson JS, Hunt JA, et al. The role of calcified cartilage and subchondral bone in the initiation and progression of ochronotic arthropathy in alkaptonuria. Arthritis Rheum. 2011;63(12):3887-96. doi: 10.1002/art.30606. [PubMed: 22127706].
6. Gil JA, Wawrzynski J, Waryasz GR. Orthopedic manifestations of ochronosis: Pathophysiology, presentation, diagnosis, and management. Am J Med. 2016;129(5):536. doi: 10.1016/j.amjmed.2016.01.010. [PubMed: 26844634].
7. Harun M, Hayrettin Y, Serhat M, Cuneyt M, Firat F, Ufuk O. A rare cause of arthropathy: An ochronotic patient with black joints. Int J Surg Case Rep. 2014;5(8):554-7. doi: 10.1016/j.ijscr.2014.06.015. [PubMed: 25034257]. [PubMed Central: PMC4147659].
8. Patel VG. Total knee arthroplasty in ochronosis. Arthroplast Today. 2015;1(3):77-80. doi: 10.1016/j.artd.2015.03.003. [PubMed: 28326376]. [PubMed Central: PMC4956685].
9. Rathore FA, Ayaz SB, Mansoor SN. Ochronotic arthropathy: Two case reports from a developing country. Clin Med Insights Arthritis Musculoskelet Disord. 2016;9:15-20. doi:
10.4137/CMAMD.S31560. [PubMed: 26884684]. [PubMed Central: PMC4749042].
10. Siavashi B, Zehtab MJ, Pendar E. Ochronosis of hip joint; A case report. Cases J. 2009;2:9337. doi: 10.1186/1757-1626-2-9337. [PubMed: 20062596]. [PubMed Central: PMC2803996].
11. Dom K, Pittevils T. Ochronotic arthropathy: the black hip. Case report and review of the literature. Acta Orthop Belg. 1997;63(2):122-5. [PubMed: 9265798].
12. Cebesoy O, Isik M, Subasi M, Kaya A, Bilgin F, Kaya O. Total hip replacement for an ochronotic patient: A technical trick. Am J Case Rep. 2014;15:27-30. doi: 10.12659/AJCR.890008. [PubMed: 24459540]. [PubMed Central: PMC3899173].
13. Aliberti G, Pulignano I, Pisani D, Rocchietti MM, Del PF, Proietta M. Bisphosphonate treatment in ochronotic osteoporotic
patients. Clin Rheumatol. 2007;26(5):729-35. doi:
10.1007/s10067-006-0390-3. [PubMed: 16924393].
14. Pachore JA, Shah VI, Upadhyay S, Shah K, Sheth A, Kshatriya A. Primary hip arthroplasty for the treatment of alkaptonuric hip arthritis: 3-to 24-year follow-ups. Arthroplasty. 2019;1(1):1-9. doi: 10.1186/s42836-019-0010-8.
15. Couto A, Sa Rodrigues A, Oliveira P, Seara M. Ochronotic arthropathy-a rare clinical case. Oxf Med Case Reports. 2018;2018(9):omy069. doi: 10.1093/omcr/omy069. [PubMed: 30174828]. [PubMed Central: PMC6112307].
16. Acar MA, Erkocak OF, Aydin BK, Altan E, Senaran H, Elmadag NM. Patients with black hip and black knee due to ochronotic arthropathy: Case report and review of literature. Oman Med J. 2013;28(6):448-9. doi: 10.5001/omj.2013.124. [PubMed: 24223251]. [PubMed Central: PMC3815858].
2. Schiavone ML, Pecorelli A, Woodby B, Ferrara F, Pambianchi E, Santucci A, et al. Mechanisms involved in the unbalanced redox homeostasis in osteoblastic cellular model of Alkaptonuria.
Arch Biochem Biophys. 2020;690:108416. doi:
10.1016/j.abb.2020.108416. [PubMed: 32502471].
3. Helliwell TR, Gallagher JA, Ranganath L. Alkaptonuria-a review of surgical and autopsy pathology. Histopathology.
2008;53(5):503-12.doi:10.1111/j.1365-2559.2008.03000.x.
[PubMed: 18336562].
4. Martin JP Jr, Batkoff B. Homogentisic acid autoxidation and oxygen radical generation: Implications for the etiology of alkaptonuric arthritis. Free Radic Biol Med. 1987;3(4):241-50. doi: 10.1016/s0891-5849(87)80031-x. [PubMed: 3121448].
5. Taylor AM, Boyde A, Wilson PJ, Jarvis JC, Davidson JS, Hunt JA, et al. The role of calcified cartilage and subchondral bone in the initiation and progression of ochronotic arthropathy in alkaptonuria. Arthritis Rheum. 2011;63(12):3887-96. doi: 10.1002/art.30606. [PubMed: 22127706].
6. Gil JA, Wawrzynski J, Waryasz GR. Orthopedic manifestations of ochronosis: Pathophysiology, presentation, diagnosis, and management. Am J Med. 2016;129(5):536. doi: 10.1016/j.amjmed.2016.01.010. [PubMed: 26844634].
7. Harun M, Hayrettin Y, Serhat M, Cuneyt M, Firat F, Ufuk O. A rare cause of arthropathy: An ochronotic patient with black joints. Int J Surg Case Rep. 2014;5(8):554-7. doi: 10.1016/j.ijscr.2014.06.015. [PubMed: 25034257]. [PubMed Central: PMC4147659].
8. Patel VG. Total knee arthroplasty in ochronosis. Arthroplast Today. 2015;1(3):77-80. doi: 10.1016/j.artd.2015.03.003. [PubMed: 28326376]. [PubMed Central: PMC4956685].
9. Rathore FA, Ayaz SB, Mansoor SN. Ochronotic arthropathy: Two case reports from a developing country. Clin Med Insights Arthritis Musculoskelet Disord. 2016;9:15-20. doi:
10.4137/CMAMD.S31560. [PubMed: 26884684]. [PubMed Central: PMC4749042].
10. Siavashi B, Zehtab MJ, Pendar E. Ochronosis of hip joint; A case report. Cases J. 2009;2:9337. doi: 10.1186/1757-1626-2-9337. [PubMed: 20062596]. [PubMed Central: PMC2803996].
11. Dom K, Pittevils T. Ochronotic arthropathy: the black hip. Case report and review of the literature. Acta Orthop Belg. 1997;63(2):122-5. [PubMed: 9265798].
12. Cebesoy O, Isik M, Subasi M, Kaya A, Bilgin F, Kaya O. Total hip replacement for an ochronotic patient: A technical trick. Am J Case Rep. 2014;15:27-30. doi: 10.12659/AJCR.890008. [PubMed: 24459540]. [PubMed Central: PMC3899173].
13. Aliberti G, Pulignano I, Pisani D, Rocchietti MM, Del PF, Proietta M. Bisphosphonate treatment in ochronotic osteoporotic
patients. Clin Rheumatol. 2007;26(5):729-35. doi:
10.1007/s10067-006-0390-3. [PubMed: 16924393].
14. Pachore JA, Shah VI, Upadhyay S, Shah K, Sheth A, Kshatriya A. Primary hip arthroplasty for the treatment of alkaptonuric hip arthritis: 3-to 24-year follow-ups. Arthroplasty. 2019;1(1):1-9. doi: 10.1186/s42836-019-0010-8.
15. Couto A, Sa Rodrigues A, Oliveira P, Seara M. Ochronotic arthropathy-a rare clinical case. Oxf Med Case Reports. 2018;2018(9):omy069. doi: 10.1093/omcr/omy069. [PubMed: 30174828]. [PubMed Central: PMC6112307].
16. Acar MA, Erkocak OF, Aydin BK, Altan E, Senaran H, Elmadag NM. Patients with black hip and black knee due to ochronotic arthropathy: Case report and review of literature. Oman Med J. 2013;28(6):448-9. doi: 10.5001/omj.2013.124. [PubMed: 24223251]. [PubMed Central: PMC3815858].
| Files | ||
| Issue | Vol 6, No 4 (2020) | |
| Section | Case Report | |
| DOI | https://doi.org/10.18502/jost.v6i4.5569 | |
| Keywords | ||
| Arthroplasty; Alkaptonuria; Ochronosis | ||
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This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License. |
How to Cite
1.
Kazemi M, Rabie H, sharafi MH. Black Femoral Head Due to Ochronotic Arthropathy: A Case Report. J Orthop Spine Trauma. 2021;6(4):108-110.
