Literature DB >> 20492825

Cutaneous mastocytosis: Two pediatric cases treated with topical pimecrolimus.

Osvaldo Correia1, Ana Filipa Duarte, Paula Quirino, Rosa Azevedo, Luis Delgado.   

Abstract

Cutaneous mastocytosis is characterized by increased numbers of skin mast cells that release mediators causing pruritus, urticaria, and flushing. Most pediatric mastocytosis patients exhibit the pattern of urticaria pigmentosa, which typically appears during the first two years of life and resolves spontaneously in late adolescence. However, while the disease is active, patients are frequently symptomatic and uncomfortable, which justifies symptomatic treatment. We report 2 patients, a 14-month-old girl and a 26-month-old boy, with localized cutaneous erythematous lesions with a positive Darier sign. In each, a punch biopsy confirmed the diagnosis of mastocytosis. Treatment was instituted with pimecrolimus cream twice a day and oral antihistamine. An almost complete response was achieved after 4 months of therapy in both patients, with no clinical evidence of recurrence after 4 years and 2 years of follow-up, respectively. In children, the treatment of mastocytosis is directed primarily to avoiding potential mast cell degranulating agents and alleviating symptoms. Topical calcineurin inhibitors act by inhibiting T-cell activation and cytokine release; they may suppress mast cell- mediated reactions by reducing their degranulation. These two cases suggest that in localized cutaneous mastocytosis they are a safe and efficacious alternative to topical steroid therapy.

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Year:  2010        PMID: 20492825

Source DB:  PubMed          Journal:  Dermatol Online J        ISSN: 1087-2108


  8 in total

Review 1.  Pediatric Mastocytosis: A Review of the Literature.

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Journal:  Pediatr Allergy Immunol Pulmonol       Date:  2013-12-01       Impact factor: 1.349

Review 2.  [Childhood-onset mastocytosis].

Authors:  F Siebenhaar; K Weller; U Blume-Peytavi; M Maurer
Journal:  Hautarzt       Date:  2012-02       Impact factor: 0.751

Review 3.  Clinical relevance of inherited genetic differences in human tryptases: Hereditary alpha-tryptasemia and beyond.

Authors:  Sarah C Glover; Melody C Carter; Peter Korošec; Patrizia Bonadonna; Lawrence B Schwartz; Joshua D Milner; George H Caughey; Dean D Metcalfe; Jonathan J Lyons
Journal:  Ann Allergy Asthma Immunol       Date:  2021-08-13       Impact factor: 6.248

Review 4.  Mastocytosis: update on pharmacotherapy and future directions.

Authors:  Juan Carlos Cardet; Cem Akin; Min Jung Lee
Journal:  Expert Opin Pharmacother       Date:  2013-10       Impact factor: 3.889

Review 5.  Mediator-Related Symptoms and Anaphylaxis in Children with Mastocytosis.

Authors:  Knut Brockow; Katarzyna Plata-Nazar; Magdalena Lange; Bogusław Nedoszytko; Marek Niedoszytko; Peter Valent
Journal:  Int J Mol Sci       Date:  2021-03-07       Impact factor: 5.923

Review 6.  Pharmacological treatment options for mast cell activation disease.

Authors:  Gerhard J Molderings; Britta Haenisch; Stefan Brettner; Jürgen Homann; Markus Menzen; Franz Ludwig Dumoulin; Jens Panse; Joseph Butterfield; Lawrence B Afrin
Journal:  Naunyn Schmiedebergs Arch Pharmacol       Date:  2016-04-30       Impact factor: 3.000

7.  Maculopapular Cutaneous Mastocytosis in a Six-month-old Boy Who Presented with Respiratory Distress.

Authors:  Maira Abdul Razzak; Laila Tul Qadar; Maria Khan; Mohammad Hasan; Ammarah Jamal
Journal:  Cureus       Date:  2019-02-22

Review 8.  Molecular Background, Clinical Features and Management of Pediatric Mastocytosis: Status 2021.

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

  8 in total

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