J Mashiah1,2, A Harel1, C Bodemer3,4,5, S Hadj-Rabia3,4,5, I Goldberg2,6, E Sprecher1,2,6, A Kutz1. 1. Pediatric Dermatology Unit, Dana Children's Hospital, Tel Aviv Sourasky Medical Center, Tel Aviv, Israel. 2. Department of Dermatology, Tel Aviv Sourasky Medical Center, Tel Aviv, Israel. 3. Department of Dermatology, Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC), Institut Imagine, Hôpital Universitaire Necker-Enfants Malades, AP-HP, Paris, France. 4. INSERM 1163, Institut Imagine, Paris, France. 5. Université Paris Descartes-Sorbonne Paris Cité, Paris, France. 6. Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Abstract
BACKGROUND: Most cases of paediatric cutaneous mastocytosis (CM) occur before the age of 2 years, and regression occurs in only 67% of children. Given the absence of any specific therapy, CM is usually treated symptomatically. A few publications have reported the beneficial effect of calcineurin inhibitors for CM. AIM: We sought to evaluate the clinical effectiveness and safety profile of topical pimecrolimus cream for the treatment of CM. METHODS: We performed a retrospective study of all diagnosed cases of CM treated with topical pimecrolimus 1% cream between 2013 and 2015. All patients were evaluated in a paediatric dermatology unit of a tertiary medical centre. Epidemiological, clinical and treatment data, including effectiveness and safety, were reviewed. RESULTS: In total, 18 children (11 male, 7 female; age range 3-42 months) with CM were evaluated. Of the 146 treated lesions, 39 (26.7%) disappeared and 98 (67%) faded or developed postinflammatory hyperpigmentation. Of the 119 papular lesions, 24 (16.4%) showed partial flattening and 56 (47%) became macular. Darier sign became negative in 14 (82%) of 17 patients. No topical or systemic complications were observed. CONCLUSIONS: Topical therapy with pimecrolimus 1% cream should be considered in the treatment of CM.
BACKGROUND: Most cases of paediatric cutaneous mastocytosis (CM) occur before the age of 2 years, and regression occurs in only 67% of children. Given the absence of any specific therapy, CM is usually treated symptomatically. A few publications have reported the beneficial effect of calcineurin inhibitors for CM. AIM: We sought to evaluate the clinical effectiveness and safety profile of topical pimecrolimus cream for the treatment of CM. METHODS: We performed a retrospective study of all diagnosed cases of CM treated with topical pimecrolimus 1% cream between 2013 and 2015. All patients were evaluated in a paediatric dermatology unit of a tertiary medical centre. Epidemiological, clinical and treatment data, including effectiveness and safety, were reviewed. RESULTS: In total, 18 children (11 male, 7 female; age range 3-42 months) with CM were evaluated. Of the 146 treated lesions, 39 (26.7%) disappeared and 98 (67%) faded or developed postinflammatory hyperpigmentation. Of the 119 papular lesions, 24 (16.4%) showed partial flattening and 56 (47%) became macular. Darier sign became negative in 14 (82%) of 17 patients. No topical or systemic complications were observed. CONCLUSIONS: Topical therapy with pimecrolimus 1% cream should be considered in the treatment of CM.
Authors: Magdalena Lange; Karin Hartmann; Melody C Carter; Frank Siebenhaar; Ivan Alvarez-Twose; Inés Torrado; Knut Brockow; Joanna Renke; Ninela Irga-Jaworska; Katarzyna Plata-Nazar; Hanna Ługowska-Umer; Justyna Czarny; Anna Belloni Fortina; Francesca Caroppo; Roman J Nowicki; Bogusław Nedoszytko; Marek Niedoszytko; Peter Valent Journal: Int J Mol Sci Date: 2021-03-04 Impact factor: 5.923