Literature DB >> 11306829

Clinico-immunological heterogeneity in Comèl-Netherton syndrome.

D Van Gysel1, H Koning, M R Baert, H F Savelkoul, H J Neijens, A P Oranje.   

Abstract

BACKGROUND: Comèl-Netherton syndrome (CN) is characterized by atopic-eczema-like skin abnormalities combined with linear ichthyotic lesions, hair shaft abnormalities and atopy with high IgE levels.
OBJECTIVE: Five children with CN are described. In 2 of the 3 CN patients still alive, analysis of cytokines regulating IgE synthesis was performed.
METHODS: In peripheral blood mononuclear cells and cultures of purified T cells, mRNA expression and protein production of interleukin 4 (IL-4), IL-13, IL-5 and interferon gamma were analysed. The results were compared with the values in age-matched atopic dermatitis patients and healthy children.
RESULTS: The 5 CN patients showed striking differences in disease severity and evolution. Marked differences were found in several cytokines in the 2 analysed CN patients. Low percentages of natural killer cells were observed in both CN patients.
CONCLUSION: The regulation of IgE production in patients with CN is varied and complex. The CN patients were heterogeneous in terms of Th2 skewing. Copyright 2001 S. Karger AG, Basel.

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Year:  2001        PMID: 11306829     DOI: 10.1159/000051607

Source DB:  PubMed          Journal:  Dermatology        ISSN: 1018-8665            Impact factor:   5.366


  10 in total

1.  Ichthyosis molecular fingerprinting shows profound TH17 skewing and a unique barrier genomic signature.

Authors:  Kunal Malik; Helen He; Thy Nhat Huynh; Gary Tran; Kelly Mueller; Kristina Doytcheva; Yael Renert-Yuval; Tali Czarnowicki; Shai Magidi; Margaret Chou; Yeriel D Estrada; Huei-Chi Wen; Xiangyu Peng; Hui Xu; Xiuzhong Zheng; James G Krueger; Amy S Paller; Emma Guttman-Yassky
Journal:  J Allergy Clin Immunol       Date:  2018-05-24       Impact factor: 10.793

Review 2.  Profiling Immune Expression to Consider Repurposing Therapeutics for the Ichthyoses.

Authors:  Amy S Paller
Journal:  J Invest Dermatol       Date:  2019-01-19       Impact factor: 8.551

3.  An IL-17-dominant immune profile is shared across the major orphan forms of ichthyosis.

Authors:  Amy S Paller; Yael Renert-Yuval; Maria Suprun; Hitokazu Esaki; Margeaux Oliva; Thy Nhat Huynh; Benjamin Ungar; Norma Kunjravia; Rivka Friedland; Xiangyu Peng; Xiuzhong Zheng; Yeriel D Estrada; James G Krueger; Keith A Choate; Mayte Suárez-Fariñas; Emma Guttman-Yassky
Journal:  J Allergy Clin Immunol       Date:  2016-08-20       Impact factor: 10.793

4.  Netherton syndrome: report of identical twins presenting with severe atopic dermatitis.

Authors:  Gurkan Kilic; Nermin Guler; Ulker Ones; Zeynep Tamay; Pinar Guzel
Journal:  Eur J Pediatr       Date:  2006-05-03       Impact factor: 3.183

5.  Comèl-Netherton syndrome defined as primary immunodeficiency.

Authors:  Ellen D Renner; Dominik Hartl; Stacey Rylaarsdam; Marguerite L Young; Linda Monaco-Shawver; Gary Kleiner; M Louise Markert; E Richard Stiehm; Bernd H Belohradsky; Melissa P Upton; Troy R Torgerson; Jordan S Orange; Hans D Ochs
Journal:  J Allergy Clin Immunol       Date:  2009-08-14       Impact factor: 10.793

6.  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

7.  Significant response to Pembrolizumab for metastatic cutaneous squamous cell carcinoma in patient with Netherton syndrome.

Authors:  Salem M Tos; Bilal Nabeel Alqam; Narmeen Giacaman; Mohammad G Ibdah; Mahmoud M M Gabajah; Abdallah Altell
Journal:  Ann Med Surg (Lond)       Date:  2022-08-03

Review 8.  Proposal for a 6-step approach for differential diagnosis of neonatal erythroderma.

Authors:  E Cuperus; A Bygum; L Boeckmann; C Bodemer; M C Bolling; M Caproni; A Diociaiuti; S Emmert; J Fischer; A Gostynski; S Guez; M E van Gijn; K Hannulla-Jouppi; C Has; A Hernández-Martín; A E Martinez; J Mazereeuw-Hautier; M Medvecz; I Neri; V Sigurdsson; K Suessmuth; H Traupe; V Oji; S G M A Pasmans
Journal:  J Eur Acad Dermatol Venereol       Date:  2022-03-15       Impact factor: 9.228

9.  Comel-Netherton syndrome: A local skin barrier defect in the absence of an underlying systemic immunodeficiency.

Authors:  Kira Stuvel; Jorn J Heeringa; Virgil A S H Dalm; Ruud W J Meijers; Els van Hoffen; Susan A M Gerritsen; Menno C van Zelm; Suzanne G M A Pasmans
Journal:  Allergy       Date:  2020-02-21       Impact factor: 13.146

10.  Dupilumab Improves Pruritus in Netherton Syndrome: A Case Study.

Authors:  Yutaka Inaba; Nobuo Kanazawa; Kyoko Muraoka; Azusa Yariyama; Ami Kawaguchi; Kayo Kunimoto; Chikako Kaminaka; Yuki Yamamoto; Kaoru Tsujioka; Akira Yoshida; Teruki Yanagi; Masatoshi Jinnin
Journal:  Children (Basel)       Date:  2022-02-24
  10 in total

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