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Case Reports
. 2023 Jan 6;11(1):210-217.
doi: 10.12998/wjcc.v11.i1.210.

Ochronotic arthropathy of bilateral hip joints: A case report

Nicolas Yap San Min  1 , Urba Rafi  1 , Juan Wang  2 , Bin He  1 , Lei Fan  3
Affiliations
Case Reports

Ochronotic arthropathy of bilateral hip joints: A case report

Nicolas Yap San Min et al. World J Clin Cases. .

Abstract

Background: Ochronosis, also known as alkaptonuria, is a rare autosomal recessive self-metabolic disease arising from deficiency of homogentisate 1,2 dioxygenase enzyme. It affects several organs and muscoskeletal structures. We herein report a case of a patient who presented with severe hip arthropathy complicated with late stage ochronosis.

Case summary: A 56-year-old male patient was admitted in our department in 2019 with complaints of chronic low backache and left hip pain. After the required investigations were done, lumbar disc herniation and severe hip arthritis were the initial diagnosis. A total left hip arthroplasty was performed. Ochronotic osteoarthritis was only obtained post-surgery as confirmatory diagnosis. He was again admitted mid 2022 with the same complaints on the right hip. Subsequently, he underwent a total right hip arthroplasty. Post-operative recovery and follow-ups were deemed very satisfactory.

Conclusion: Ochronosis is an unusual diagnosis for a patient who presents with typical hip arthritis. Thus, unless meticulous history taking and advanced laboratory tests, the diagnosis can easily be missed by surgeons.

Keywords: Alkaptonuria; Bilateral hip arthroplasty; Case report; Hip arthritis; Metabolic disease; Ochronotic osteoarthropathy.

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Conflict of interest statement

Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.

Figures

Figure 1
Figure 1
X-rays of lumbar spine and left hip. A and B: Degenerative changes, lumbar vertebra spondylitis of lumbar spine; C: Pelvis indicated bilateral hip arthritis, aseptic necrosis of left hip joint of lumbar spine; D: Anteroposterior view of left hip; E: Lateral view showed disappearance of hip space and aseptic necrosis (stage 4) of left femoral head of left hip.
Figure 2
Figure 2
Magnetic resonance imaging of both hips. A: Aseptic necrosis of the left femoral head and left hip arthritis; B: Edema of right femoral head and cervical bone marrow; C: Bilateral acetabular cystic ischemia; D: Edema in the left gluteus maximus intermuscular space.
Figure 3
Figure 3
During second admission in July 2022, pelvis X-ray indicated a marked degeneration of the right hip. No joint space seen, loss of shape of the right femoral head.
Figure 4
Figure 4
Right femoral head during right total hip arthroplasty. Deformed femoral head, black and brown deposits were seen similarly as compared to his left femoral head.
Figure 5
Figure 5
Post-operative right hip X-ray. A: Anteroposterior view; B: Lateral view showed satisfactory prosthesis placement.
Figure 6
Figure 6
During surgery, the femoral head was black with a large amount of black and brown material deposits.
Figure 7
Figure 7
Post-operative right hip X-ray. A: Anteroposterior view; B: Lateral view showed satisfactory prosthesis placement.
Figure 8
Figure 8
Urine 24 h test. A: Morning urine after collection; B: Brownish yellow urine after 24 h rest.
Figure 9
Figure 9
Eye examination revealed yellow-brown plaques in both sclerae. A: Right eye; B: Left eye.
See this image and copyright information in PMC

References

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