Thierry Passeron1, Aicha Salhi2, Jean-Michel Mazer3, Céline Lavogiez4, Juliette Mazereeuw-Hautier5, Chrystèle Galliot6, Anne-Marie Collet-Villette7, Christine Labreze8, Laurence Boon9, Jean-Philippe Hardy9, Virginie Fayard6, Cristina Bulai Livideanu5, Gérard Toubel10, Gabriela Georgescou11, Nathalie Gral12, Aude Maza5, Jean-Philippe Lacour13. 1. Department of Dermatology of Nice, University Hospital of Nice, Nice, France; Institut National de Santé et de Recherche Médicale (INSERM) U1065, Team 12, C3M, Nice, France. Electronic address: passeron@unice.fr. 2. Department of Dermatology of Alger, University Hospital of Alger, Alger, Algeria. 3. Centre Laser International de la Peau, Paris, France. 4. Department of Dermatology of Lille, University Hospital of Lille, Lille, France. 5. Department of Dermatology of Toulouse, University Hospital of Toulouse, Toulouse, France. 6. Laser Center, Paris, France. 7. Department of Dermatology of Marseille, University Hospital of Marseille, Marseille, France. 8. Department of Dermatology of Bordeaux, University Hospital of Bordeaux, Bordeaux, France. 9. Center for Vascular Anomalies, Division of Plastic Surgery, Cliniques Universitaires Saint-Luc, Brussels, Belgium. 10. Laser Center, Rennes, France. 11. Department of Dermatology of Tours, University Hospital of Tours, Tours, France. 12. Laser Center, Grenoble, France. 13. Department of Dermatology of Nice, University Hospital of Nice, Nice, France.
Abstract
BACKGROUND: There is limited information regarding early development of soft-tissue and/or bone hypertrophy with facial port-wine stains (PWS). OBJECTIVE: We sought to characterize patients with hypertrophic PWS presenting during childhood. METHODS: Patients with a facial PWS and underlying hypertrophy that developed before the age of 18 years were included in a multicenter retrospective study. Age at onset of the hypertrophy, its location, association with odontologic problems, presence of other associated complications, and response to laser treatment were recorded. RESULTS: A total of 98 patients were included. The mean age at onset of hypertrophy, retrieved for 77 of 98 patients, was 5.6 years. The hypertrophy was congenital in 26%. Odontologic problems were noted in 39.8% of cases. Other complications, including cataract, asymmetric development of the maxillary bone, and speech delay/disorders, were reported in 18.4%. In all, 67 patients received laser treatment. Only 3% achieved complete or nearly complete clearance of the PWS. LIMITATIONS: As only cases of PWS with early-onset hypertrophy were included, we were unable to calculate the prevalence of this manifestation. CONCLUSION: PWS with early-onset hypertrophy are associated with a high rate of complications and a poor response to laser treatment. Periodic monitoring is recommended for early detection and treatment of complications.
BACKGROUND: There is limited information regarding early development of soft-tissue and/or bone hypertrophy with facial port-wine stains (PWS). OBJECTIVE: We sought to characterize patients with hypertrophic PWS presenting during childhood. METHODS:Patients with a facial PWS and underlying hypertrophy that developed before the age of 18 years were included in a multicenter retrospective study. Age at onset of the hypertrophy, its location, association with odontologic problems, presence of other associated complications, and response to laser treatment were recorded. RESULTS: A total of 98 patients were included. The mean age at onset of hypertrophy, retrieved for 77 of 98 patients, was 5.6 years. The hypertrophy was congenital in 26%. Odontologic problems were noted in 39.8% of cases. Other complications, including cataract, asymmetric development of the maxillary bone, and speech delay/disorders, were reported in 18.4%. In all, 67 patients received laser treatment. Only 3% achieved complete or nearly complete clearance of the PWS. LIMITATIONS: As only cases of PWS with early-onset hypertrophy were included, we were unable to calculate the prevalence of this manifestation. CONCLUSION:PWS with early-onset hypertrophy are associated with a high rate of complications and a poor response to laser treatment. Periodic monitoring is recommended for early detection and treatment of complications.
Authors: Sophie E R Horbach; Albert Wolkerstorfer; Daniel Martijn de Bruin; Sanne M Jansen; Chantal M A M van der Horst Journal: BMJ Open Date: 2017-11-14 Impact factor: 2.692