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Background: Ankylosing spondylitis (AS) is a chronic rheumatic disease that primarily affects the spine and the sacroiliac and peripheral joints. Juvenile-onset AS (JoAS) patients will likely present with peripheral joint symptoms. Knee flexion contracture (KFC) and hip flexion contracture (HFC) are common in these patients due to subchondral bone inflammation. The Ilizarov technique is the most commonly used technique for treating KFC. However, its use to treat JoAS-associated KFC has not been reported., Case Summary: This report presents a case study of a 31-year-old male patient with a squatting gait due to severe bilateral KFC and HFC. The patient had a normal walking pattern until the age of eight, after which he experienced knee and hip pain, leading to the gradual development of KFC and HFC. The patient's primary complaint was an inability to walk upright. The patient was diagnosed with JoAS and underwent hip dissection and release, limited soft tissue release of the hamstring, and gradual traction using the Ilizarov method. Ultimately, the patient was able to walk upright., Conclusion: The incidence of squatting gait due to KFC in individuals diagnosed with JoAS was low. Utilizing the Ilizarov technique has proven to be a secure and effective method for managing KFC in JoAS patients. Although the Ilizarov technique cannot substitute for total knee arthroplasty (TKA), its application can delay the need for primary TKA in JoAS patients and alleviate the intricacy and potential complications associated with the procedure., Competing Interests: Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article., (©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.)