Literature DB >> 35322332

Separating gene clustering in the rare mucopolysaccharidosis disease.

Leon Bobrowski1,2, Tomasz Łukaszuk1, Lidia Gaffke3, Zuzanna Cyske3, Mariusz Ferenc2, Karolina Pierzynowska3, Grzegorz Węgrzyn4.   

Abstract

Rare disease datasets are typically structured such that a small number of patients (cases) are represented by multidimensional feature vectors. In this report, we considered a rare disease, mucopolysaccharidosis (MPS). This disease is divided into 11 types and subtypes, depending on the genetic defect, type of deficient enzyme, and nature of accumulated glycosaminoglycan(s). Among them, 7 types are known as possibly neuronopathic and 4 are non-neuronopathic, and in the case of the former group, prediction of the course of the disease is crucial for patient's treatment and the management. Here, we have used transcriptomic data available for one patient from each MPS type/subtype. The approach to gene grouping considered by us was based on the minimization of the perceptron criterion in the form of convex and piecewise linear function (CPL). This approach allows designing complexes of linear classifiers on the basis of small samples of multivariate vectors. As a result, distinguishing neuronopathic and non-neuronopathic forms of MPS was possible on the basis of bioinformatic analysis of gene expression patterns where each MPS type was represented by only one patient. This approach can be potentially used also for assessing other features of patients suffering from rare diseases, for which large body of data (like transcriptomic data) is available from only one or a few representatives.
© 2022. The Author(s), under exclusive licence to Institute of Plant Genetics Polish Academy of Sciences.

Entities:  

Keywords:  Complexes of linear classifiers; Data mining; Gene clustering; Perceptron criterion function; Rare diseases

Mesh:

Year:  2022        PMID: 35322332     DOI: 10.1007/s13353-022-00691-2

Source DB:  PubMed          Journal:  J Appl Genet        ISSN: 1234-1983            Impact factor:   3.240


  5 in total

1.  Mutation spectrum and pivotal features for differential diagnosis of Mucopolysaccharidosis IVA patients with severe and attenuated phenotype.

Authors:  Beyhan Tüysüz; Dilek Uludağ Alkaya; Güven Toksoy; Nilay Güneş; Timur Yıldırım; İlhan Avni Bayhan; Zehra Oya Uyguner
Journal:  Gene       Date:  2019-04-11       Impact factor: 3.688

2.  Correlation between severity of mucopolysaccharidoses and combination of the residual enzyme activity and efficiency of glycosaminoglycan synthesis.

Authors:  Ewa Piotrowska; Joanna Jakóbkiewicz-Banecka; Anna Tylki-Szymańska; Barbara Czartoryska; Alicja Wegrzyn; Grzegorz Wegrzyn
Journal:  Acta Paediatr       Date:  2008-11-30       Impact factor: 2.299

3.  The mutational spectrum of hunter syndrome reveals correlation between biochemical and clinical profiles in Tunisian patients.

Authors:  L Chkioua; O Grissa; N Leban; M Gribaa; H Boudabous; H Ben Turkia; S Ferchichi; N Tebib; S Laradi
Journal:  BMC Med Genet       Date:  2020-05-24       Impact factor: 2.103

4.  Genotype-phenotype relationships in mucopolysaccharidosis type I (MPS I): Insights from the International MPS I Registry.

Authors:  Lorne A Clarke; Roberto Giugliani; Nathalie Guffon; Simon A Jones; Hillary A Keenan; Maria V Munoz-Rojas; Torayuki Okuyama; David Viskochil; Chester B Whitley; Frits A Wijburg; Joseph Muenzer
Journal:  Clin Genet       Date:  2019-07-02       Impact factor: 4.438

5.  A cDNA analysis disclosed the discordance of genotype-phenotype correlation in a patient with attenuated MPS II and a 76-base deletion in the gene for iduronate-2-sulfatase.

Authors:  Yasuyuki Fukuhara; Ai Miura; Narutoshi Yamazaki; Tetsumin So; Motomichi Kosuga; Kumiko Yanagi; Tadashi Kaname; Takanori Yamagata; Hitoshi Sakuraba; Torayuki Okuyama
Journal:  Mol Genet Metab Rep       Date:  2020-12-10
  5 in total

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