Literature DB >> 31656314

Alazami syndrome: the first case of papillary thyroid carcinoma.

Ivan Ivanovski1,2, Stefano Giuseppe Caraffi1, Elisa Magnani3, Simonetta Rosato1, Marzia Pollazzon1, Leslie Matalonga4, Simonetta Piana5, Davide Nicoli6, Chiara Baldo7, Sergio Bernasconi8, Andrea Frasoldati3, Orsetta Zuffardi9, Livia Garavelli10.   

Abstract

Alazami syndrome (MIM#615071) is a rare developmental disorder caused by biallelic variants in the LARP7 gene. Hallmark features include short stature, global developmental delay, and distinctive facial features. To date, 23 patients from 11 families have been reported in the literature. Here we describe a 19-year-old man who, in association with the typical features of Alazami syndrome, was diagnosed at the age of 14 years with papillary thyroid carcinoma, harboring the somatic BRAF V600E mutation. Whole exome sequencing revealed two novel LARP7 variants in compound heterozygosity, whereas only common variants were detected in genes associated with familial nonmedullary thyroid cancer (MIM#188550). LARP7 acts as a tumor suppressor in breast and gastric cancer, and possibly, according to recent studies, in thyroid tumors. Since thyroid cancer is rare among children and adolescents, we hypothesize that the LARP7 variants identified in our patient are responsible for both Alazami syndrome and tumor susceptibility. We also provide an overview of the clinical findings in all Alazami syndrome patients reported to date and discuss the possible pathogenetic mechanism that may underlie this condition, including the role of LARP7 in tumor susceptibility.

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Year:  2019        PMID: 31656314     DOI: 10.1038/s10038-019-0682-5

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  3 in total

1.  Updating the neurodevelopmental profile of Alazami syndrome: Illustrating the role of developmental assessment in rare genetic disorders.

Authors:  Monica H Wojcik; Kate Linnea; Joan M Stoler; Leonard Rappaport
Journal:  Am J Med Genet A       Date:  2019-05-10       Impact factor: 2.802

2.  hLARP7 C-terminal domain contains an xRRM that binds the 3' hairpin of 7SK RNA.

Authors:  Catherine D Eichhorn; Rahul Chug; Juli Feigon
Journal:  Nucleic Acids Res       Date:  2016-09-26       Impact factor: 16.971

3.  LARP7 in papillary thyroid carcinoma induces NIS expression through suppression of the SHH signaling pathway.

Authors:  Xiaomei Sui; Yana Sui; Yonghui Wang
Journal:  Mol Med Rep       Date:  2018-04-05       Impact factor: 2.952

  3 in total
  4 in total

1.  Stabilize and connect: the role of LARP7 in nuclear non-coding RNA metabolism.

Authors:  Daniele Hasler; Gunter Meister; Utz Fischer
Journal:  RNA Biol       Date:  2020-06-03       Impact factor: 4.652

2.  The RD-Connect Genome-Phenome Analysis Platform: Accelerating diagnosis, research, and gene discovery for rare diseases.

Authors:  Steven Laurie; Davide Piscia; Leslie Matalonga; Alberto Corvó; Marcos Fernández-Callejo; Carles Garcia-Linares; Carles Hernandez-Ferrer; Cristina Luengo; Inés Martínez; Anastasios Papakonstantinou; Daniel Picó-Amador; Joan Protasio; Rachel Thompson; Raul Tonda; Mònica Bayés; Gemma Bullich; Jordi Camps-Puchadas; Ida Paramonov; Jean-Rémi Trotta; Angel Alonso; Marcella Attimonelli; Christophe Béroud; Virginie Bros-Facer; Orion J Buske; Andrés Cañada-Pallarés; José M Fernández; Mats G Hansson; Rita Horvath; Julius O B Jacobsen; Rajaram Kaliyaperumal; Séverine Lair-Préterre; Luana Licata; Pedro Lopes; Estrella López-Martín; Deborah Mascalzoni; Lucia Monaco; Luis A Pérez-Jurado; Manuel Posada de la Paz; Jordi Rambla; Ana Rath; Olaf Riess; Peter N Robinson; David Salgado; Damian Smedley; Dylan Spalding; Peter A C 't Hoen; Ana Töpf; Irina Zaharieva; Holm Graessner; Ivo G Gut; Hanns Lochmüller; Sergi Beltran
Journal:  Hum Mutat       Date:  2022-06       Impact factor: 4.700

3.  Compound Phenotype Due to Recessive Variants in LARP7 and OTOG Genes Disclosed by an Integrated Approach of SNP-Array and Whole Exome Sequencing.

Authors:  Pietro Palumbo; Orazio Palumbo; Maria Pia Leone; Ester di Muro; Stefano Castellana; Luigi Bisceglia; Tommaso Mazza; Massimo Carella; Marco Castori
Journal:  Genes (Basel)       Date:  2020-03-31       Impact factor: 4.096

4.  Identification of NID1 as a novel candidate susceptibility gene for familial non-medullary thyroid carcinoma using whole-exome sequencing.

Authors:  Luis Eduardo Barbalho de Mello; Thaise Nayane Ribeiro Carneiro; Aline Neves Araujo; Camila Xavier Alves; Pedro Alexandre Favoretto Galante; Vanessa Candiotti Buzatto; Maria das Graças de Almeida; Karina Marques Vermeulen-Serpa; Sancha Helena de Lima Vale; Fernando José de Pinto Paiva; José Brandão-Neto; Janete Maria Cerutti
Journal:  Endocr Connect       Date:  2022-01-31       Impact factor: 3.335

  4 in total

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