Multisystemic Manifestations in Rare Diseases: The Experience of Dyskeratosis Congenita.


Journal

Genes
ISSN: 2073-4425
Titre abrégé: Genes (Basel)
Pays: Switzerland
ID NLM: 101551097

Informations de publication

Date de publication:
11 03 2022
Historique:
received: 29 11 2021
revised: 07 03 2022
accepted: 08 03 2022
entrez: 25 3 2022
pubmed: 26 3 2022
medline: 27 4 2022
Statut: epublish

Résumé

Dyskeratosis congenital (DC) is the first genetic syndrome described among telomeropathies. Its classical phenotype is characterized by the mucocutaneous triad of reticulated pigmentation of skin lace, nail dystrophy and oral leukoplakia. The clinical presentation, however, is heterogeneous and serious clinical complications include bone marrow failure, hematological and solid tumors. It may also involve immunodeficiencies, dental, pulmonary and liver disorders, and other minor complication. Dyskeratosis congenita shows marked genetic heterogeneity, as at least 14 genes are responsible for the shortening of telomeres characteristic of this disease. This review discusses clinical characteristics, molecular genetics, disease evolution, available therapeutic options and differential diagnosis of dyskeratosis congenita to provide an interdisciplinary and personalized medical assessment that includes family genetic counseling.

Identifiants

pubmed: 35328050
pii: genes13030496
doi: 10.3390/genes13030496
pmc: PMC8953471
pii:
doi:

Types de publication

Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

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Auteurs

Michele Callea (M)

Pediatric Dentistry and Special Dental Care Unit, Meyer Children's University Hospital, 50139 Florence, Italy.

Diego Martinelli (D)

Unit of Metabolism, Bambino Gesù Children's Research Hospital, Piazza Sant'Onofrio, 4, 00165 Rome, Italy.

Francisco Cammarata-Scalisi (F)

Servicio de Pediatría, Hospital Regional de Antofagasta, Antofagasta 1240835, Chile.

Chiara Grimaldi (C)

Department of Pediatric Surgery, Meyer Children's Hospital, Viale Gaetano Pieraccini 24, 50139 Florence, Italy.

Houweyda Jilani (H)

Genetic Department, Mongi Slim Hospital, Marsa 2046, Tunisia.
Faculty of Medicine of Tunis, University of Tunis El Manar, Tunis 1068, Tunisia.

Piercesare Grimaldi (P)

Department of Public Health and Pediatric Sciences, University of Torino, 10125 Torino, Italy.

Colin Eric Willoughby (CE)

Genomic Medicine, Biomedical Sciences Research Institute, Ulster University, Coleraine Campus, Coleraine BT52 1SA, UK.

Antonino Morabito (A)

Department of Pediatric Surgery, Meyer Children's Hospital, Viale Gaetano Pieraccini 24, 50139 Florence, Italy.
Department of Neurofarba, University of Florence, Viale Pieraccini 6, 50121 Florence, Italy.

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