Literature DB >> 27190931

The Arid Melancholy-Netherton Syndrome With Protein Energy Malnutrition.

Sonia Pramod Jain1, Pramod Ajit Jain2, Neha Pandey3.   

Abstract

Netherton Syndrome (NS) is a rare autosomal recessive hereditary ichthyosiform disease with a classical triad comprising of an ichthyosiform dermatosis, hair shaft abnormalities and atopic diathesis. There is a mutation in a gene named Serine Protease Inhibitor Kazal type-5 (SPINK5); a new type of serine protease inhibitor involved in the regulation of skin barrier formation and immunity. Skin manifestations include, Ichthyosis Linearis Circumflexa (ILC), polycyclic and serpiginous, erythematous plaques with characteristic migratory, double-edged scale at the margins, or Congenital Ichthyosiform Erythroderma (CIE). Most of the patients have elevated immunoglobulin class E (IgE) and show atopic manifestations. Hair shaft abnormalities like pili torti and/or trichorrhexis nodosa, trichorrhexis invaginata, are seen. Here, we report a rare case of Netherton Syndrome having ILC and trichorrhexis nodosa with protein energy malnutrition in a five-year-old school going girl. She belonged to a poor socio-economic background and was worried about her physical appearance due to her skin lesions, causing psychosocial morbidity to her.

Entities:  

Keywords:  Ichthyosis linearis circumflexa; LEKTI; SPINK5; Trichorrhexis nodosa

Year:  2016        PMID: 27190931      PMCID: PMC4866229          DOI: 10.7860/JCDR/2016/18200.7655

Source DB:  PubMed          Journal:  J Clin Diagn Res        ISSN: 0973-709X


  6 in total

1.  Netherton syndrome: disease expression and spectrum of SPINK5 mutations in 21 families.

Authors:  Emmanuelle Bitoun; Stéphane Chavanas; Alan D Irvine; Lorne Lonie; Christine Bodemer; Mauro Paradisi; Dominique Hamel-Teillac; Shin-ichi Ansai; Yoshihiko Mitsuhashi; Alain Taïeb; Yves de Prost; Giovanna Zambruno; John I Harper; Alain Hovnanian
Journal:  J Invest Dermatol       Date:  2002-02       Impact factor: 8.551

2.  Ichthyosis linearis circumflexa.

Authors:  S Malakar; K Lahiri; S R Sengupta
Journal:  Indian J Dermatol Venereol Leprol       Date:  1996 Nov-Dec       Impact factor: 2.545

3.  Netherton syndrome with pili torti.

Authors:  Sahana M Srinivas; Ravi Hiremagalore; Swetha Suryanarayan; Leelavathy Budamakuntala
Journal:  Int J Trichology       Date:  2013-10

4.  Bamboo Hair in Netherton's Syndrome.

Authors:  Atul D Salodkar; Sanjiv V Choudhary; Gori Jadwani; Adarshlata Singh
Journal:  Int J Trichology       Date:  2009-07

5.  Netherton syndrome in one Chinese adult with a novel mutation in the SPINK5 gene and immunohistochemical studies of LEKTI.

Authors:  Zhang Xi-Bao; Zhang San-Quan; He Yu-Qing; Luo Yu-Wu; Luo Quan; Li Chang-Xing
Journal:  Indian J Dermatol       Date:  2012-07       Impact factor: 1.494

6.  Netherton syndrome, a case report and review of literature.

Authors:  Hassan Boskabadi; Gholamali Maamouri; Shahin Mafinejad
Journal:  Iran J Pediatr       Date:  2013-10       Impact factor: 0.364

  6 in total

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