Literature DB >> 33525735

Gene Expression Analysis of Mevalonate Kinase Deficiency Affected Children Identifies Molecular Signatures Related to Hematopoiesis.

Simona Pisanti1, Marianna Citro1, Mario Abate1, Mariella Caputo1, Rosanna Martinelli1.   

Abstract

Mevalonate kinase deficiency (MKD) is a rare autoinflammatory genetic disorder characterized by recurrent fever attacks and systemic inflammation with potentially severe complications. Although it is recognized that the lack of protein prenylation consequent to mevalonate pathway blockade drives IL1β hypersecretion, and hence autoinflammation, MKD pathogenesis and the molecular mechanisms underlaying most of its clinical manifestations are still largely unknown. In this study, we performed a comprehensive bioinformatic analysis of a microarray dataset of MKD patients, using gene ontology and Ingenuity Pathway Analysis (IPA) tools, in order to identify the most significant differentially expressed genes and infer their predicted relationships into biological processes, pathways, and networks. We found that hematopoiesis linked biological functions and pathways are predominant in the gene ontology of differentially expressed genes in MKD, in line with the observed clinical feature of anemia. We also provided novel information about the molecular mechanisms at the basis of the hematological abnormalities observed, that are linked to the chronic inflammation and to defective prenylation. Considering the broad and unspecific spectrum of MKD clinical manifestations and the difficulty in its diagnosis, a better understanding of MKD molecular bases could be translated to the clinical level to facilitate diagnosis, and improve management and therapy.

Entities:  

Keywords:  anemia; hematopoiesis; mevalonate kinase deficiency; stress erythropoiesis

Year:  2021        PMID: 33525735      PMCID: PMC7908123          DOI: 10.3390/ijerph18031170

Source DB:  PubMed          Journal:  Int J Environ Res Public Health        ISSN: 1660-4601            Impact factor:   3.390


  45 in total

1.  Loss of a Rho-regulated actin nucleator, mDia2, impairs cytokinesis during mouse fetal erythropoiesis.

Authors:  Sadanori Watanabe; Tihana De Zan; Toshimasa Ishizaki; Shingo Yasuda; Hiroshi Kamijo; Daisuke Yamada; Tomohiro Aoki; Hiroshi Kiyonari; Hiroshi Kaneko; Ritsuko Shimizu; Masayuki Yamamoto; Gohta Goshima; Shuh Narumiya
Journal:  Cell Rep       Date:  2013-11-14       Impact factor: 9.423

Review 2.  A Comprehensive Overview of the Hereditary Periodic Fever Syndromes.

Authors:  Donato Rigante; Bruno Frediani; Luca Cantarini
Journal:  Clin Rev Allergy Immunol       Date:  2018-06       Impact factor: 8.667

Review 3.  Rho GTPases in erythroid maturation.

Authors:  Theodosia A Kalfa; Yi Zheng
Journal:  Curr Opin Hematol       Date:  2014-05       Impact factor: 3.284

Review 4.  Mevalonate kinase deficiency (hyper IgD syndrome with periodic fever)--different faces with separate treatments: two cases and review of the literature.

Authors:  Pınar Gençpınar; Balahan B Makay; Marco Gattorno; Francesco Caroli; Erbil Ünsal
Journal:  Turk J Pediatr       Date:  2012 Nov-Dec       Impact factor: 0.552

5.  Suppression of Fas-FasL coexpression by erythropoietin mediates erythroblast expansion during the erythropoietic stress response in vivo.

Authors:  Ying Liu; Ramona Pop; Cameron Sadegh; Carlo Brugnara; Volker H Haase; Merav Socolovsky
Journal:  Blood       Date:  2006-03-09       Impact factor: 22.113

6.  Reappraising the role of α5 integrin and the microenvironmental support in stress erythropoiesis.

Authors:  Tatyana Ulyanova; Grigorios Georgolopoulos; Thalia Papayannopoulou
Journal:  Exp Hematol       Date:  2019-12-28       Impact factor: 3.084

7.  The Phenotype and Genotype of Mevalonate Kinase Deficiency: A Series of 114 Cases From the Eurofever Registry.

Authors:  Nienke M Ter Haar; Jerold Jeyaratnam; Helen J Lachmann; Anna Simon; Paul A Brogan; Matteo Doglio; Marco Cattalini; Jordi Anton; Consuelo Modesto; Pierre Quartier; Esther Hoppenreijs; Silvana Martino; Antonella Insalaco; Luca Cantarini; Loredana Lepore; Maria Alessio; Inmaculada Calvo Penades; Christina Boros; Rita Consolini; Donato Rigante; Ricardo Russo; Jana Pachlopnik Schmid; Thirusha Lane; Alberto Martini; Nicolino Ruperto; Joost Frenkel; Marco Gattorno
Journal:  Arthritis Rheumatol       Date:  2016-11       Impact factor: 10.995

Review 8.  Management of Mevalonate Kinase Deficiency: A Pediatric Perspective.

Authors:  Jerold Jeyaratnam; Joost Frenkel
Journal:  Front Immunol       Date:  2020-06-05       Impact factor: 7.561

Review 9.  Mevalonate kinase deficiency: current perspectives.

Authors:  Leslie A Favier; Grant S Schulert
Journal:  Appl Clin Genet       Date:  2016-07-20

Review 10.  Stress Erythropoiesis is a Key Inflammatory Response.

Authors:  Robert F Paulson; Baiye Ruan; Siyang Hao; Yuanting Chen
Journal:  Cells       Date:  2020-03-06       Impact factor: 6.600

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.