Patient-Oriented Priorities for Pediatric Erythromelalgia: A Priority-Setting Process.
Children (Basel) 2025 Nov 02; 12(11).

Abstract

Background/Objectives
: 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.
Methods
: 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.
Results
: 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.
Conclusions
: 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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Authors+Show Affiliations

Ocay DD0000-0003-4265-8647Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital, Boston, MA 02115, USA. Department of Anaesthesia, Harvard Medical School, Boston, MA 02115, USA.
Halpin M0000-0002-7589-071XDepartment of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital, Boston, MA 02115, USA.
Ford EPatient/Parent Partner.
Keighley KPatient/Parent Partner.
Keighley NPatient/Parent Partner.
Ramsay NPatient/Parent Partner.
Ramsay TPatient/Parent Partner.
Sheridan CMPatient/Parent Partner.
Sheridan SMPatient/Parent Partner.
Tice KRPatient/Parent Partner.
De Ranieri D0000-0003-0782-6000Division of Rheumatology, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL 60611, USA. Department of Pediatrics, Feinberg School of Medicine, Northwestern University, Chicago, IL 60611, USA.
Tham SWDepartment of Anesthesiology and Pain Medicine, School of Medicine, University of Washington, Seattle, WA 98105, USA. Department of Anesthesiology and Pain Medicine, Seattle Children's Hospital, Seattle, WA 98105, USA.
Brownstein CA0000-0002-7371-0340Division of Genetics and Genomics, Department of Pediatrics, Boston Children's Hospital, Boston, MA 02115, USA. Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA.
Clinch JDepartment of Pediatric Rheumatology, Bristol Royal Hospital for Children, Bristol BS2 8BJ, UK.
Davis DM0000-0001-6819-6044Department of Dermatology, Mayo Clinic Rochester, Rochester, MN 55905, USA. Department of Pediatric and Adolescent Medicine, Mayo Clinic Rochester, Rochester, MN 55905, USA.
Donado C0000-0003-4345-9201Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital, Boston, MA 02115, USA. Department of Anaesthesia, Harvard Medical School, Boston, MA 02115, USA.
D'Souza G0000-0003-4801-6215Department of Anesthesiology, Perioperative and Pain Medicine, Stanford University, Stanford, CA 94305, USA.
Kattail D0000-0002-8388-4192Department of Anesthesia and Pain Medicine, The Hospital for Sick Children (SickKids), Toronto, ON M5G 1X8, Canada.
Lobo K0000-0003-4328-4894Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital, Boston, MA 02115, USA.
Ravetti DPatient/Parent Partner.
Sandroni PDepartment of Neurology, Mayo Clinic Rochester, Rochester, MN 55905, USA.
Stinson JN0000-0002-9969-8052Department of Anesthesia and Pain Medicine, The Hospital for Sick Children (SickKids), Toronto, ON M5G 1X8, Canada. Research Institute, The Hospital for Sick Children (SickKids), Toronto, ON M5G 1X8, Canada.
Walco GA0000-0003-0812-0210Department of Anesthesiology and Pain Medicine, School of Medicine, University of Washington, Seattle, WA 98105, USA. Department of Anesthesiology and Pain Medicine, Seattle Children's Hospital, Seattle, WA 98105, USA.
Walker SM0000-0002-6086-9459Developmental Neurosciences, University College London Great Ormond Street Institute of Child Health, London WC1N 1EH, UK. Department of Paediatric Anaesthesia and Pain Medicine, Great Ormond Street Hospital NHS Foundation Trust, London WC1N 3BH, UK.
Yu TWDivision of Genetics and Genomics, Department of Pediatrics, Boston Children's Hospital, Boston, MA 02115, USA. Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA.
Berde CBDepartment of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital, Boston, MA 02115, USA. Department of Anaesthesia, Harvard Medical School, Boston, MA 02115, USA.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

41300595