• Ana Cristina Paredes, Pedro Teixeira, Armando Almeida, and Patrícia Ribeiro Pinto.
• Life and Health Sciences Research Institute (ICVS), School of Medicine, University of Minho, Braga, Portugal; ICVS/3B's - PT Government Associate Laboratory, Braga/Guimarães, Portugal.
• J Pain. 2021 Oct 1; 22 (10): 113411451134-1145.

AbstractChronic pain is a common condition among people with hemophilia (PWH), associated with joint deterioration due to repeated joint bleeds. This systematic review and meta-analysis aimed to determine the prevalence of chronic pain due to haemophilia and to analyze its interference in the lives of patients. A systematic search was performed in May and June 2019 and updated in February 2021, using PubMed, EMBASE, Web of Science and SciElo. The search included terms related to hemophilia, pain, pain prevalence and pain interference. Studies were included if they reported data referring to hemophilia-related chronic pain among adult males (age ≥18). From 3,258 identified studies, 11 met the inclusion criteria. Three studies used a proposed definition for hemophilia-related chronic pain and 8 used direct questions developed by the authors. For the global samples, prevalence ranged from 17% to 84%. The random-effects meta-analysis including all studies demonstrated a pooled prevalence of chronic pain of 46% (95% Confidence Interval, CI = 34%-58%). Subgroup analysis of samples including all disease severities or including only severe patients revealed a pooled prevalence of 48% (95% CI = 29%-67%) and 53% (95% CI = 38%-69%), respectively. High heterogeneity between studies was observed in all models. Information concerning chronic pain interference was retrieved from 1 study, reporting a mean interference of 3.7 (0-10 numerical rating scale from the Brief Pain Inventory). This systematic review revealed a wide prevalence range of hemophilia-related chronic pain across studies, varying methodologies and sample characteristics. Research in the hemophilia field should clearly distinguish between acute and chronic pain and provide complete characterization of study samples. PERSPECTIVE: Pain is a central issue in the lives of people with hemophilia, posing a significant challenge for healthcare providers. A clear picture of chronic pain due to hemophilia is precluded by high heterogeneity among studies and various definitions used to investigate its prevalence.Copyright © 2021 United States Association for the Study of Pain, Inc. Published by Elsevier Inc. All rights reserved.

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