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Original Investigation |

Risk Factors for Degree and Type of Sequelae After Involution of Untreated Hemangiomas of Infancy ONLINE FIRST

Eulalia Baselga, MD1; Esther Roe, MD1; Julien Coulie, MD2; Fania Z. Muñoz, MD1; Laurence M. Boon, MD2; Catherine McCuaig, MD3; Angela Hernandez-Martín, MD4; Ignasi Gich, MD5; Luis Puig, MD1
[+] Author Affiliations
1Department of Dermatology, Hospital de la Santa Creu i Sant Pau, Universitat Autònoma de Barcelona, Barcelona, Spain
2Center for Vascular Anomalies, Division of Plastic Surgery, Cliniques Universitaires St Luc, Brussels, Belgium
3Division of Dermatology (Pediatrics) CHU Sainte-Justine, University of Montreal, Montreal, Canada
4Department of Dermatology, Hospital Infantil Universitario del Niño Jesús, Madrid, Spain
5Department of Clinical Epidemiology; CIM-Caiber-IIb Sant Pau, Barcelona Spain
JAMA Dermatol. Published online August 17, 2016. doi:10.1001/jamadermatol.2016.2905
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Importance  Infantile hemangiomas involute to some extent, but they often leave sequelae that may cause disfigurement. Factors determining the risk of permanent sequelae after regression are of crucial importance in treatment decision making.

Objectives  To describe the sequelae left by infantile hemangiomas after natural involution and to identify clinical characteristics that could predict the most severe or a particular type of sequelae.

Design, Setting, and Participants  Multicentric retrospective cohort study of images from 187 infantile hemangiomas that had not received systemic treatment and had follow-up pictures until regression that were selected from photographic files taken between 2003 and 2013 at 4 university hospitals with large vascular clinics in 3 different countries.

Main Outcomes and Measures  Outcome measures were the type of sequelae classified as residual telangiectasia, anetodermal skin, redundant skin, persistent superficial component, and the degree of sequelae ranging from 1 to 4.

Results  A total of 184 hemangiomas were included. The overall incidence of significant sequelae was 101 of 184 (54.9%). The most common sequelae after involution were telangiectasias (145, 84.3%), fibrofatty tissue (81, 47.1%), and anetodermic skin (56, 32.6%). The average age at which hemangioma completed involution was 3.5 years. Superficial and deep hemangiomas left significantly fewer sequelae than combined hemangiomas (Mann-Whitney; superficial vs deep, OR, 1.6; 95% CI, 0.6-3.8; P = .81; superficial vs combined, OR, 3.3; 95% CI, 1.7-6.3; P < .001; deep vs combined, OR, 2.1; 95% CI, 0.9-5.1; P < .001). Hemangiomas with a step or abrupt border of the superficial component left more severe sequelae than those with a smooth border (χ2,OR, 3.4; 95% CI, 1.8-6.6; P < .001). Superficial hemangiomas with a cobblestone appearance or rough surface left more severe sequelae than those with a smooth surface (Kruskal-Wallis; α, 0.05; P < .001). Using multivariate analysis, combined hemangiomas with a superficial component and a step border were associated with more sequelae.

Conclusions and Relevance  In this retrospective study of sequelae in a large cohort of untreated infants, we quantified the prevalence of permanent scarring and identified clinical features predictive of permanent sequelae. Our observations provide useful information at a time when the treatment paradigm for hemangiomas has changed. Such knowledge may help primary care physicians predict the risk of sequelae and identify high-risk lesions to implement early treatment.

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Figure 1.
Examples of Different Types of Sequelae

A, Deep hemangioma that regressed without sequelae; B, superficial hemangioma that left only telangiectasia; C, mixed hemangioma that left anetodermic skin; D, mixed hemangioma that left redundant skin; and E, mixed hemangioma that left fibrofatty tissue.

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Figure 2.
Comparison of Sequelae Associated With Different Borders for the Superficial Component

A, Patient 21 at age 8 months (left), and age 3 years (right); B, patient 8 at age 8 months (left) and 3 years (right). Superficial hemangioma with a step border (patient 21, A) left more sequelae than a mixed hemangioma of the same size and location but with a progressive border (patient 8, B).

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Figure 3.
Comparison of Sequelae Associated With Different Surface Features of the Superficial Component

A, Patient 22 at age 6 months (left) and age 4 years (right); B, patient 31 at age 8 months (left) and 2 years (right). Hemangiomas with a smooth surface leave less sequelae (patient 22, A), than hemangiomas with a cobblestoned surface (patient 31, B).

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