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Chronic pain is a common experience in osteogenesis imperfecta (OI). However, there are few studies on this topic, and none of them emerge from psychology as a discipline. The purpose of this work is to describe the frequency of chronic pain and its characteristics in a large sample of adults with OI, as well as its relationship with clinical, sociodemographic, psychological, and quality of life variables. A cross-sectional study was carried out in a sample of 418 adults with OI who answered a battery of online questionnaires. Sociodemographic and clinical variables, pain parameters, participants' appraisal of pain, coping strategies, interference in daily activities, and health-related quality of life were evaluated. A descriptive and correlational analysis was performed. Up to 83% of the sample reported experiencing pain frequently. Both the frequency and intensity of pain were related to the accumulation of fractures over the years (P < .05), but were independent of other variables like the severity of the pathology or the use of bisphosphonates. Higher threat appraisal of pain was associated with an increase in perceived pain intensity and its interference with daily activities, as well as a decrease in physical and mental health (P < .001). Chronic pain frequent condition in adults with OI, regardless of the severity of the pathology. It interferes with their usual activities and has an impact on their quality of life. The way in which participants appraise their pain also have an influence on its intensity and its consequences. Interventions aimed at training strategies for managing appraisals about pain could potentially improve adaptation to chronic pain., Competing Interests: The authors have no conflicts of interest to disclose., (Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.)