Objectives: Infantile hemangiomas (IHs) are common; some cases require timely referral and treatment to prevent complications. We developed and validated a reliable instrument for timely and adequate referral of patients with IH to experts by nonexpert primary physicians.
Methods: In this multicenter, cross-sectional, observational study, we used a 3-stage process: (1) development of the Infantile Hemangioma Referral Score (IHReS) tool by IH experts who selected a representative set of 42 IH cases comprising images and a short clinical history, (2) definition of the gold standard for the 42 cases by a second independent committee of IH experts, and (3) IHReS validation by nonexpert primary physicians using the 42 gold standard cases.
Results: A total of 60 primary physicians from 7 different countries evaluated the 42 gold standard cases (without reference to the IHReS tool); 45 primary physicians evaluated these cases using the IHReS questionnaire, and 44 completed retesting using the instrument. IHReS had a sensitivity of 96.9% (95% confidence interval 96.1%-97.8%) and a specificity of 55.0% (95% confidence interval 51.0%-59.0%). The positive predictive value and negative predictive value were 40.5% and 98.3%, respectively. Validation by experts and primary physicians revealed substantial agreement for interrater reliability and intrarater repeatability.
Conclusions: IHReS, a 2-part algorithm with a total of 12 questions, is an easy-to-use tool for primary physicians for the purpose of facilitating correct and timely referral of patients with IH. IHReS may help practitioners in their decision to refer patients to expert centers.
Copyright © 2020 by the American Academy of Pediatrics.
Conflict of interest statement
POTENTIAL CONFLICT OF INTEREST: Dr Léauté-Labrèze is a consultant and clinical trial investigator for Pierre Fabre and is a member of the Infantile Hemangioma European Task Force (IHETF) and received consultant fees. Dr Baselga Torres is a consultant and speaker for Pierre Fabre, is a member of the IHETF and received consultant fees, is a consultant for Ferrer and Sanofi, is the founder and consultant for Vanthera, and is a speaker for Almirall. Dr Weibel is member of the IHETF and received consultant fees; she is a consultant and speaker for Sanofi, MEDA, Merz, Permamed, and La Roche Posay (L’Oréal) and the principal investigator in clinical trials with AbbVie, Novartis, Eli Lilly, and Pfizer. Dr Boon is member of the IHETF and received consultant fees and has relationships with the following companies: Deciphera Pharmaceuticals, Inc (Waltham, MA), Venthera, Inc (Palo Alto, CA), and Pfizer, Inc (Ixelles, Belgium) (sirolimus or VASE study). Dr El Hachem is member of the IHETF and received consultant fees. Dr van der Vleuten is a member of the IHETF and received consultant fees. Dr Roessler is a consultant for Pierre Fabre, is a member of the IHETF and received consultant fees, is a consultant for SOBI, and has received research grants from Pfizer and travel grants from Roche. Dr Troilius Rubin is a member of the IHETF and received consultant fees.
Hemangioma Activity and Severity Index (HASI), an instrument for evaluating infantile hemangioma: development and preliminary validation.Int J Dermatol. 2015 Apr;54(4):494-8. doi: 10.1111/ijd.12646. Epub 2014 Dec 29. Int J Dermatol. 2015. PMID: 25557642
Measuring the severity of infantile hemangiomas: instrument development and reliability.Arch Dermatol. 2012 Feb;148(2):197-202. doi: 10.1001/archdermatol.2011.926. Arch Dermatol. 2012. PMID: 22351819
Burden of Infantile Hemangioma on Family: An International Observational Cross-Sectional Study.Pediatr Dermatol. 2017 May;34(3):295-302. doi: 10.1111/pde.13133. Epub 2017 Apr 6. Pediatr Dermatol. 2017. PMID: 28382698 Clinical Trial.
Infantile hemangiomas, complications and treatments.Semin Cutan Med Surg. 2016 Sep;35(3):108-16. doi: 10.12788/j.sder.2016.050. Semin Cutan Med Surg. 2016. PMID: 27607318 Review.
Clinical and economic impact of surgery for treating infantile hemangiomas in the era of propranolol: overview of single-center experience from La Paz Hospital, Madrid.Eur J Pediatr. 2019 Jan;178(1):1-6. doi: 10.1007/s00431-018-3290-z. Epub 2018 Nov 12. Eur J Pediatr. 2019. PMID: 30421264 Review.
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