A Tunisian family with a novel mutation in the gene CYP4F22 for lamellar ichthyosis and co-occurrence of hearing loss in a child due to mutation in the SLC26A4 gene.


Journal

International journal of dermatology
ISSN: 1365-4632
Titre abrégé: Int J Dermatol
Pays: England
ID NLM: 0243704

Informations de publication

Date de publication:
Dec 2019
Historique:
received: 28 06 2018
revised: 06 03 2019
accepted: 11 03 2019
pubmed: 26 4 2019
medline: 6 5 2020
entrez: 26 4 2019
Statut: ppublish

Résumé

Co-occurrence of two genetic diseases is challenging for accurate diagnosis and genetic counseling. The recent availability of whole exome sequencing (WES) has dramatically improved the molecular diagnosis of rare genetic diseases in particular in consanguineous populations. We report here on a consanguineous family from Southern Tunisia including three members affected with congenital ichthyosis. The index case had a hearing loss (HL) and ichthyosis and was primarily suspected as suffering from keratitis-ichthyosis-deafness (KID) syndrome. WES was performed for the index case, and all members of the nuclear family were sequenced (Sanger method). The WES approach allowed the identification of two strong candidate variants in two different genes; a missense mutation c.1334T>G (p.Leu445Trp) in exon 11 of SLC26A4 gene, associated with isolated HL and a novel missense mutation c.728G>T (p.Arg243Leu) in exon 8 of CYP4F22 gene likely responsible for ichthyosis. These two mutations were predicted to be pathogenic by three pathogenicity prediction softwares (Scale-Invariant Feature Transform [SIFT], Polymorphism Phenotyping [PolyPhen], Mutation Taster) to underlie the HL and ichthyosis, respectively. The present study raises awareness about the importance of familial history for accurate diagnosis of syndromic genetic diseases and differential diagnosis with co-occurrence of two distinct clinical entities. In addition, in countries with limited resources, WES sequencing for a single individual provides a cost effective tool for molecular diagnosis confirmation and genetic counseling.

Sections du résumé

BACKGROUND BACKGROUND
Co-occurrence of two genetic diseases is challenging for accurate diagnosis and genetic counseling. The recent availability of whole exome sequencing (WES) has dramatically improved the molecular diagnosis of rare genetic diseases in particular in consanguineous populations.
METHODS METHODS
We report here on a consanguineous family from Southern Tunisia including three members affected with congenital ichthyosis. The index case had a hearing loss (HL) and ichthyosis and was primarily suspected as suffering from keratitis-ichthyosis-deafness (KID) syndrome. WES was performed for the index case, and all members of the nuclear family were sequenced (Sanger method).
RESULTS RESULTS
The WES approach allowed the identification of two strong candidate variants in two different genes; a missense mutation c.1334T>G (p.Leu445Trp) in exon 11 of SLC26A4 gene, associated with isolated HL and a novel missense mutation c.728G>T (p.Arg243Leu) in exon 8 of CYP4F22 gene likely responsible for ichthyosis. These two mutations were predicted to be pathogenic by three pathogenicity prediction softwares (Scale-Invariant Feature Transform [SIFT], Polymorphism Phenotyping [PolyPhen], Mutation Taster) to underlie the HL and ichthyosis, respectively.
CONCLUSIONS CONCLUSIONS
The present study raises awareness about the importance of familial history for accurate diagnosis of syndromic genetic diseases and differential diagnosis with co-occurrence of two distinct clinical entities. In addition, in countries with limited resources, WES sequencing for a single individual provides a cost effective tool for molecular diagnosis confirmation and genetic counseling.

Identifiants

pubmed: 31020658
doi: 10.1111/ijd.14452
doi:

Substances chimiques

SLC26A4 protein, human 0
Sulfate Transporters 0
Cytochrome P-450 Enzyme System 9035-51-2
CYP4F22 protein, human EC 1.-

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1439-1443

Subventions

Organisme : Tunisian Ministry of Public Health
Organisme : Ministry of Higher Education and Scientific Research
ID : LR11IPT05
Organisme : E.C.
ID : 295097

Informations de copyright

© 2019 The International Society of Dermatology.

Références

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Auteurs

Marwa Sayeb (M)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.

Zied Riahi (Z)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.
INSERM, UMRS 1120, Vision Institut, Paris, France.
University of Paris VI UPMC Sorbonnes, Paris, France.

Nadia Laroussi (N)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.

Crystel Bonnet (C)

INSERM, UMRS 1120, Vision Institut, Paris, France.
University of Paris VI UPMC Sorbonnes, Paris, France.

Lilia Romdhane (L)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.
Department of Biology, Faculty of Science of Bizerte, Université Tunis Carthage, Zarzouna, Tunisia.

Rahma Mkaouar (R)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.

Anissa Zaouak (A)

Department of Dermatology, Habib Thameur Hospital, Tunis, Tunisia.

Jihene Marrakchi (J)

Department of Otorhinolaryngology, CHU La Rabta, Tunis, Tunisia.

Ghaith Abdessalem (G)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.

Olfa Messaoud (O)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.

Oussema Bouchniba (O)

Departement of Biochemistry, Habib Bourguiba Hospital, Medenine, Tunisia.

Nacer Ghilane (N)

Departement of Biochemistry, Habib Bourguiba Hospital, Medenine, Tunisia.

Mourad Mokni (M)

Department of Dermatology, CHU La Rabta Tunis, Tunis, Tunisia.

Ghazi Besbes (G)

Department of Otorhinolaryngology, CHU La Rabta, Tunis, Tunisia.

Houda Yacoub-Youssef (H)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.

Christine Petit (C)

INSERM, UMRS 1120, Vision Institut, Paris, France.
University of Paris VI UPMC Sorbonnes, Paris, France.
Unité de Génétique et Physiologie de l'Audition, Institut Pasteur, Paris, France.
Collège de France, Paris, France.

Sonia Abdelhak (S)

Laboratory of Biomedical Genomics and Oncogenetics (LR11IPT05), University of Tunis El Manar, Pasteur Institut of Tunis, Tunis, Tunisia.

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