Literature DB >> 31195081

Profound immunodeficiency with severe skin disease explained by concomitant novel CARMIL2 and PLEC1 loss-of-function mutations.

Maria E Maccari1, Carsten Speckmann1, Maximilian Heeg1, Antonia Reimer2, Federica Casetti2, Cristina Has2, Stephan Ehl1, Carla N Castro3.   

Abstract

This study reports a patient with severe skin disease in the context of profound immunodeficiency explained by two concomitant genetic diseases caused by two novel homozygous loss-of-function mutations in PLEC1 and CARMIL2. The work provides additional information on the clinical and immunological manifestations of CARMIL2 deficiency and highlights the particular diagnostic and therapeutic challenge represented by the concomitant presence of two rare monogenic disorders.
Copyright © 2019 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  CARMIL2; Epidermolysis bullosa; Immunodeficiency; Plectin; RLTPR; Whole exome sequencing

Mesh:

Substances:

Year:  2019        PMID: 31195081     DOI: 10.1016/j.clim.2019.06.004

Source DB:  PubMed          Journal:  Clin Immunol        ISSN: 1521-6616            Impact factor:   3.969


  6 in total

1.  CARMIL2-related immunodeficiency manifesting with photosensitivity.

Authors:  Leila H Shayegan; Maria C Garzon; Kimberly D Morel; Rachel Borlack; Patricia M Vuguin; Kara G Margolis; Yesim Y Demirdag; Elaine M Pereira; Christine T Lauren
Journal:  Pediatr Dermatol       Date:  2020-04-27       Impact factor: 1.588

Review 2.  Human genetic dissection of papillomavirus-driven diseases: new insight into their pathogenesis.

Authors:  Vivien Béziat
Journal:  Hum Genet       Date:  2020-05-20       Impact factor: 4.132

3.  Exogenous interleukin-2 can rescue in-vitro T cell activation and proliferation in patients with a novel capping protein regulator and myosin 1 linker 2 mutation.

Authors:  O Toker; M Berger; O Shamriz; A J Simon; A Lev; O Megged; O Ledder; E Picard; L Joseph; V Molho-Pessach; Y Tal; P Millman; M Slae; R Somech
Journal:  Clin Exp Immunol       Date:  2020-04-18       Impact factor: 4.330

Review 4.  Molecular Therapeutics in Development for Epidermolysis Bullosa: Update 2020.

Authors:  Cristina Has; Andrew South; Jouni Uitto
Journal:  Mol Diagn Ther       Date:  2020-06       Impact factor: 4.074

5.  A Novel Pathogenic Variant in CARMIL2 (RLTPR) Causing CARMIL2 Deficiency and EBV-Associated Smooth Muscle Tumors.

Authors:  Jennifer R Yonkof; Ajay Gupta; Cesar M Rueda; Shamlal Mangray; Benjamin T Prince; Hemalatha G Rangarajan; Mohammad Alshahrani; Elizabeth Varga; Timothy P Cripe; Roshini S Abraham
Journal:  Front Immunol       Date:  2020-06-18       Impact factor: 7.561

6.  Bronchopleural fistula in a 5- years old child with novel CARMIL 2 mutation: A rare disease and a rare case.

Authors:  Ikram Ul Haq Chaudhry; Ahmed Alshaer; Burair Al Jassas; Amal Alkhunizi; Mohammad Alsaiary; Tasneem A AlMubayaedh; Abass A AlMalki; Ahmed Almesfer
Journal:  Ann Med Surg (Lond)       Date:  2021-06-06
  6 in total

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