Patient-Oriented Priorities for Pediatric Erythromelalgia: A Priority-Setting Process

<b>Background/Objectives</b>: Erythromelalgia is a rare condition characterized by burning pain, redness, and warmth primarily in the extremities, usually worsened by heat and alleviated by cold. The objective of this study was to identify the top 10 priorities in pediatric erythromelalgia from multiple perspectives, including clinicians, people with lived experience of childhood-onset erythromelalgia, and their family members. <b>Methods</b>: A modified James Lind Alliance Priority-Setting Process was conducted. The top priorities were identified through four phases: (1) an international online survey to gather priorities, (2) data processing, (3) an interim prioritization online survey, and (4) a virtual workshop to set the final priorities. <b>Results</b>: In phase 1, 185 potential priorities were submitted by 74 respondents (53% patients, 24% family members, and 23% clinicians) that were developed into 68 unique research questions (phase 2). In phase 3, of the 68 questions, 50 were rated for importance by 58 participants (38% patients, 36% family members, and 26% clinicians), reducing the list to 25 questions. In phase 4, the top 10 was reached through consensus by 12 participants (33% patients, 25% family members, and 42% clinicians) across Canada, South Africa, the United States of America, and the United Kingdom. <b>Conclusions</b>: The final priorities focused on the treatment of erythromelalgia, understanding underlying mechanisms, the association of erythromelalgia with various body systems, and generating awareness. This list is the first international patient-centered research agenda for childhood-onset erythromelalgia and a call to action from key partners to improve future research and care.

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Publication Details

Journal
Children
Published
2025-11-02
DOI
https://doi.org/10.3390/children12111477
Primary Topic
Pain Mechanisms and Treatments
Type
article
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article

Patient-Oriented Priorities for Pediatric Erythromelalgia: A Priority-Setting Process

Travis Ramsay, Cormac Sheridan, Gary A. Walco, Meghan Halpin et al.
Children
Pain Mechanisms and Treatments
article

Patient-Oriented Priorities for Pediatric Erythromelalgia: A Priority-Setting Process

Travis Ramsay, Cormac Sheridan, Gary A. Walco, Meghan Halpin, Deepa Kattail, Dawn Marie R. Davis, Catherine A. Brownstein, See Wan Tham, Don Daniel Ocay, Suellen M. Walker, Charles B. Berde, Sarah Sheridan, Timothy W. Yu, Emily Ford, Paola Sandroni, Kimberly Lobo, Carolina Donado, Jennifer Nan Stinson, Deirdre De Ranieri, Genevieve D’Souza, Norma K.C. Ramsay, Jacqui Clinch, Danielle Ravetti, Kirsten R. Tice, Karen Keighley, Neva Keighley
article en

Abstract

<b>Background/Objectives</b>: Erythromelalgia is a rare condition characterized by burning pain, redness, and warmth primarily in the extremities, usually worsened by heat and alleviated by cold. The objective of this study was to identify the top 10 priorities in pediatric erythromelalgia from multiple perspectives, including clinicians, people with lived experience of childhood-onset erythromelalgia, and their family members. <b>Methods</b>: A modified James Lind Alliance Priority-Setting Process was conducted. The top priorities were identified through four phases: (1) an international online survey to gather priorities, (2) data processing, (3) an interim prioritization online survey, and (4) a virtual workshop to set the final priorities. <b>Results</b>: In phase 1, 185 potential priorities were submitted by 74 respondents (53% patients, 24% family members, and 23% clinicians) that were developed into 68 unique research questions (phase 2). In phase 3, of the 68 questions, 50 were rated for importance by 58 participants (38% patients, 36% family members, and 26% clinicians), reducing the list to 25 questions. In phase 4, the top 10 was reached through consensus by 12 participants (33% patients, 25% family members, and 42% clinicians) across Canada, South Africa, the United States of America, and the United Kingdom. <b>Conclusions</b>: The final priorities focused on the treatment of erythromelalgia, understanding underlying mechanisms, the association of erythromelalgia with various body systems, and generating awareness. This list is the first international patient-centered research agenda for childhood-onset erythromelalgia and a call to action from key partners to improve future research and care.

ChildrenVol. 12(11)
Northwestern University (US), Boston Children's Hospital (US), Seattle Children's Hospital (US), Mayo Clinic (US), Harvard University (US), University of Washington (US), Great Ormond Street Hospital (GB), Great Ormond Street Hospital for Children NHS Foundation Trust (GB), Hospital for Sick Children (CA), Bristol Royal Hospital for Children (GB), Mayo Clinic in Arizona (US), Lurie Children's Hospital (US), Institute of Child Health (IN), SickKids Foundation (CA), Mayo Clinic in Florida (US), University College London (GB), National Patient Safety Foundation (US), Stanford University (US)
Openalex Percentile: Top 60%
Pain Mechanisms and Treatments
DOIOpen Access PDFOpenAlex
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