Literature DB >> 32767583

A novel SPINK5 mutation and successful subcutaneous immunoglobulin replacement therapy in a child with Netherton syndrome.

Maria Zelieskova1, Peter Banovcin1, Marek Kozar2, Andrea Kozarova3, Zuzana Nudzajova4, Milos Jesenak1.   

Abstract

We report a 2-year-old patient with Netherton syndrome presenting with generalized exfoliative erythroderma, ichthyosiform dermatitis, trichorrhexis invaginata, hypernatremic dehydration, failure to thrive, and recurrent respiratory infections. Molecular analysis of SPINK5 identified a novel mutation (c.1530CA). Our case report also verifies and supports the safety and efficacy of subcutaneous immunoglobulin substitution in chronic generalized skin disorders associated with primary immunodeficiencies such as Netherton syndrome.
© 2020 Wiley Periodicals LLC.

Entities:  

Keywords:  erythroderma; genetic diseases/mechanisms; ichthyosis; immunodeficiency

Mesh:

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Year:  2020        PMID: 32767583     DOI: 10.1111/pde.14318

Source DB:  PubMed          Journal:  Pediatr Dermatol        ISSN: 0736-8046            Impact factor:   1.588


  4 in total

1.  A novel mutation in SPINK5 gene underlies a case of atypical Netherton syndrome.

Authors:  Yu Wang; Hanqing Song; Lingling Yu; Nan Wu; Xiaodong Zheng; Bo Liang; Peiguang Wang
Journal:  Front Genet       Date:  2022-09-09       Impact factor: 4.772

2.  A novel SPINK5 donor splice site variant in a child with Netherton syndrome.

Authors:  Dillon Mintoff; Isabella Borg; Julia Vornweg; Liam Mercieca; Rijad Merdzanic; Johannes Numrich; Susan Aquilina; Nikolai Paul Pace; Judith Fischer
Journal:  Mol Genet Genomic Med       Date:  2021-02-03       Impact factor: 2.183

3.  Netherton syndrome caused by compound heterozygous mutation, c.80A>G mutation in SPINK5 and large-sized genomic deletion mutation, and successful treatment of intravenous immunoglobulin.

Authors:  Zhen Zhang; Chaolan Pan; Ruoqu Wei; Huaguo Li; Yijun Yang; Jiawen Chen; Ming Li; Zhirong Yao
Journal:  Mol Genet Genomic Med       Date:  2021-01-16       Impact factor: 2.183

4.  Outcomes of Systemic Treatment in Children and Adults With Netherton Syndrome: A Systematic Review.

Authors:  Anouk E M Nouwen; Renske Schappin; N Tan Nguyen; Aviël Ragamin; Anette Bygum; Christine Bodemer; Virgil A S H Dalm; Suzanne G M A Pasmans
Journal:  Front Immunol       Date:  2022-03-30       Impact factor: 8.786

  4 in total

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