Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jan 6, 2023; 11(1): 210-217
Published online Jan 6, 2023. doi: 10.12998/wjcc.v11.i1.210
Ochronotic arthropathy of bilateral hip joints: A case report
Nicolas Yap San Min, Urba Rafi, Juan Wang, Bin He, Lei Fan
Nicolas Yap San Min, Urba Rafi, Bin He, Lei Fan, Department of Orthopedics, The Second Affiliated Hospital of Nanjing Medical University, Nanjing 210011, Jiangsu Province, China
Juan Wang, Department of Ultrasonography, The Second Affiliated Hospital of Nanjing Medical University, Nanjing 210011, Jiangsu Province, China
Author contributions: Yap San Min N and Rafi U are co-first authors and have contributed to this work equally; Yap San Min N, Rafi U and Wang J contributed to manuscript writing and editing and data collection; He B contributed to data analysis; Fan L contributed to conceptualization and supervision; and all authors have read and approved the final manuscript.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Lei Fan, MBBS, MD, PhD, Assistant Professor, Deputy Director, Doctor, Surgeon, Department of Orthopedics, The Second Affiliated Hospital of Nanjing Medical University, No. 121 Jiangjiayuan, Gulou District, Nanjing 210011, Jiangsu Province, China. fanlei8839@126.com
Received: October 10, 2022
Peer-review started: October 10, 2022
First decision: November 11, 2022
Revised: November 25, 2022
Accepted: December 21, 2022
Article in press: December 21, 2022
Published online: January 6, 2023
Processing time: 86 Days and 14.6 Hours
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; Metabolic disease; Ochronotic osteoarthropathy; Hip arthritis; Bilateral hip arthroplasty; Case report

Core Tip: Ochronosis is not an usual diagnosis in orthopedics. Thus, unless meticulous history taking and advanced laboratory tests, the diagnosis can easily be missed by surgeons. Advanced testing should come in mind when odd physical examinations are noted and especially with peculiar radiological findings. Early diagnosis and a good communication between the two parties are primordial to obtain the best prognosis.