Literature DB >> 32705701

Filling in the gaps on FILS syndrome: A case report and literature review.

Chelsea Eason1, Ahmad Aleisa1,2, Julie R Jones3, Eloise J Prijoles3, Lara Wine Lee1.   

Abstract

FILS syndrome (facial dysmorphism, immunodeficiency, livedo, and short stature) is a rare autosomal recessive disorder caused by pathogenic alterations in the POLE gene leading to multisystemic manifestations, including poorly characterized skin findings. We report a child with a homozygous variant, c.100C > T (p.Arg34Cys), in POLE and features consistent with poikiloderma, expanding the dermatologic signs associated with this rare disorder. Additionally, we review reported cases of FILS syndrome, discuss possible pathomechanisms for our patient's presentation, and consider implications for management.
© 2020 Wiley Periodicals LLC.

Entities:  

Keywords:  developmental defects; genetic diseases/mechanisms; immunodeficiency; photosensitivity

Mesh:

Year:  2020        PMID: 32705701     DOI: 10.1111/pde.14274

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


  2 in total

Review 1.  Congenital Diseases of DNA Replication: Clinical Phenotypes and Molecular Mechanisms.

Authors:  Megan Schmit; Anja-Katrin Bielinsky
Journal:  Int J Mol Sci       Date:  2021-01-18       Impact factor: 5.923

2.  Case report: A Chinese boy with facial dysmorphism, immunodeficiency, livedo, and short stature syndrome.

Authors:  Lihong Jiang; Xin Chen; Jiaqi Zheng; Meilin Wang; Hui Bo; Geli Liu
Journal:  Front Pediatr       Date:  2022-08-22       Impact factor: 3.569

  2 in total

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