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. 2023 Aug;30(3):283-287.
doi: 10.11005/jbm.2023.30.3.283. Epub 2023 Aug 31.

Black Bone Disease: Ochronotic Arthritis Detected during Knee Arthroplasty

Ho Won Kang  1 , Minsub Kim  1 , Jin-Young Oh  2 , Changhyun Youn  2
Affiliations

Black Bone Disease: Ochronotic Arthritis Detected during Knee Arthroplasty

Ho Won Kang et al. J Bone Metab. 2023 Aug.

Abstract

Alkaptonuria is an extremely rare autosomal recessive metabolic disorder characterized by dark urine, ochronosis, and arthritis of the spine and major joints. We report a case of ochronotic arthritis observed during total knee replacement surgery in a 65-year-old male patient with no relevant medical history. Based on a literature review, this is the first case of ochronotic arthritis reported in Korea.

Keywords: Alkaptonuria; Arthritis; Arthroplasty; Knee; Ochronosis.

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Conflict of interest statement

Conflict of interest

No potential conflict of interest relevant to this article was reported.

Figures

Fig. 1
Fig. 1
Bluish-black pigmentation on the skin is observed.
Fig. 2
Fig. 2
Arthritic changes in the right knee as observed on the pre-operative radiograph (A) and T2-weighted magnetic resonance image (B).
Fig. 3
Fig. 3
Black discolouration detected in the subchondral bone (A) and cancellous bone (B).
Fig. 4
Fig. 4
Post-operative anteroposterior and lateral radiographs of the right knee.
Fig. 5
Fig. 5
In alkaptonuria, mutations in the gene coding for homogentisic acid dioxygenase cause accumulation of homogentisic acid, leading to pigmentation in the connective tissue.
None
See this image and copyright information in PMC

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References

    1. Kitahara Y, Kaku N, Tagomori H, et al. Alkaptonuria with rapidly destructive arthropathy of the hip: a case report and literature review. Acta Orthop Traumatol Turc. 2021;55:563–8. doi: 10.5152/j.aott.2021.21205. - DOI - PMC - PubMed
    1. Hamdi N, Cooke TD, Hassan B. Ochronotic arthropathy: case report and review of the literature. Int Orthop. 1999;23:122–5. doi: 10.1007/s002640050325. - DOI - PMC - PubMed
    1. Keller JM, Macaulay W, Nercessian OA, et al. New developments in ochronosis: review of the literature. Rheumatol Int. 2005;25:81–5. doi: 10.1007/s00296-004-0498-1. - DOI - PubMed
    1. Rajkumar N, Soundarrajan D, Dhanasekararaja P, et al. Clinical and radiological outcomes of total joint arthroplasty in patients with ochronotic arthropathy. Eur J Orthop Surg Traumatol. 2020;30:923–9. doi: 10.1007/s00590-020-02651-0. - DOI - PubMed
    1. Shaikh A, Desai M, Kantanavar R, et al. Intraoperative diagnosis of a rare case of arthropathy - A case report and review of literature. J Orthop Case Rep. 2020;10:58–62. doi: 10.13107/jocr.2020.v10.i08.1860. - DOI - PMC - PubMed