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Comprehensive overview of disease models for Wolfram syndrome: toward effective treatments.
Morikawa S, Tanabe K, Kaneko N, Hishimura N, Nakamura A. Morikawa S, et al. Mamm Genome. 2024 Mar;35(1):1-12. doi: 10.1007/s00335-023-10028-x. Epub 2024 Feb 13. Mamm Genome. 2024. PMID: 38351344 Review.
To bridge these knowledge gaps, disease models for Wolfram syndrome are indispensable, and understanding the characteristics of each model is critical. This review will provide a summary of the current knowledge regarding WFS1 function and offer a comp …
To bridge these knowledge gaps, disease models for Wolfram syndrome are indispensable, and understanding the cha …
Wolfram syndrome: new pathophysiological insights and therapeutic strategies.
Mishra R, Chen BS, Richa P, Yu-Wai-Man P. Mishra R, et al. Ther Adv Rare Dis. 2021 Aug 16;2:26330040211039518. doi: 10.1177/26330040211039518. eCollection 2021 Jan-Dec. Ther Adv Rare Dis. 2021. PMID: 37181110 Free PMC article. Review.
Wolfram Syndrome (WS) is an ultra-rare, progressive neurodegenerative disease characterized by early-onset diabetes mellitus and irreversible loss of vision, secondary to optic nerve degeneration. ...The current landscape of therapeutic options will also be d …
Wolfram Syndrome (WS) is an ultra-rare, progressive neurodegenerative disease characterized by early-onset diabetes mel …
Endoplasmic Reticulum (ER) Stress and Endocrine Disorders.
Ariyasu D, Yoshida H, Hasegawa Y. Ariyasu D, et al. Int J Mol Sci. 2017 Feb 11;18(2):382. doi: 10.3390/ijms18020382. Int J Mol Sci. 2017. PMID: 28208663 Free PMC article. Review.
In addition to diabetes mellitus, which is well characterized, several relatively rare genetic disorders such as familial neurohypophyseal diabetes insipidus (FNDI), Wolfram syndrome, and isolated growth hormone deficiency type II (IGHD2) are discussed in this artic …
In addition to diabetes mellitus, which is well characterized, several relatively rare genetic disorders such as familial neurohypophyseal d …
Use of Zebrafish Models to Boost Research in Rare Genetic Diseases.
Crouzier L, Richard EM, Sourbron J, Lagae L, Maurice T, Delprat B. Crouzier L, et al. Int J Mol Sci. 2021 Dec 12;22(24):13356. doi: 10.3390/ijms222413356. Int J Mol Sci. 2021. PMID: 34948153 Free PMC article. Review.
In this review, we emphasize the unique advantages of zebrafish models over other in vivo models, particularly underlining the high throughput phenotypic capacity for therapeutic screening. ...Two prototypic examples of rare diseases illustrate how zebrafish mode …
In this review, we emphasize the unique advantages of zebrafish models over other in vivo models, particularly underlining the …
Evidence for altered neurodevelopment and neurodegeneration in Wolfram syndrome using longitudinal morphometry.
Lugar HM, Koller JM, Rutlin J, Eisenstein SA, Neyman O, Narayanan A, Chen L, Shimony JS, Hershey T. Lugar HM, et al. Sci Rep. 2019 Apr 12;9(1):6010. doi: 10.1038/s41598-019-42447-9. Sci Rep. 2019. PMID: 30979932 Free PMC article.
Wolfram syndrome is a rare disease caused by mutations in the WFS1 gene leading to symptoms in early to mid-childhood. ...These patterns suggest that there may be early, stalled white matter development in Wolfram syndrome, with additional degen …
Wolfram syndrome is a rare disease caused by mutations in the WFS1 gene leading to symptoms in early to mid-childhood. …
Mechanisms of pancreatic beta-cell apoptosis in diabetes and its therapies.
Johnson JD, Luciani DS. Johnson JD, et al. Adv Exp Med Biol. 2010;654:447-62. doi: 10.1007/978-90-481-3271-3_19. Adv Exp Med Biol. 2010. PMID: 20217509 Review.
There are also several rare forms of diabetes, including Wolcott-Rallison syndrome, Wolfram syndrome, as well as some forms of maturity onset diabetes of the young that are caused by mutations in genes that may play important roles in beta-cell survival. ...
There are also several rare forms of diabetes, including Wolcott-Rallison syndrome, Wolfram syndrome, as well as some f …
Hippocampus and Hypothalamus RNA-sequencing of WFS1-deficient Mice.
Ivask M, Pajusalu S, Reimann E, Kõks S. Ivask M, et al. Neuroscience. 2018 Mar 15;374:91-103. doi: 10.1016/j.neuroscience.2018.01.039. Epub 2018 Feb 3. Neuroscience. 2018. PMID: 29406269
Wolfram syndrome is caused by mutations in the WFS1 gene. WFS1 protein dysfunction results in a range of neuroendocrine syndromes and is mostly characterized by juvenile-onset diabetes mellitus and optic atrophy. ...The functional analysis revealed significant enric …
Wolfram syndrome is caused by mutations in the WFS1 gene. WFS1 protein dysfunction results in a range of neuroendocrine syndro …
Behavioral characterization of a novel Cisd2 mutant mouse.
Gaines CH, Snyder AE, Ervin RB, Farrington J, Walsh K, Schoenrock SA, Tarantino LM. Gaines CH, et al. Behav Brain Res. 2021 May 7;405:113187. doi: 10.1016/j.bbr.2021.113187. Epub 2021 Feb 18. Behav Brain Res. 2021. PMID: 33610659 Free PMC article.
Wolfram syndrome (WFS) is a rare autosomal recessive disorder characterized by diabetes mellitus and insipidus, progressive optic atrophy and sensorineural deafness. ...
Wolfram syndrome (WFS) is a rare autosomal recessive disorder characterized by diabetes mellitus and insipidus, progressive op …
Calpain inhibitor and ibudilast rescue beta cell functions in a cellular model of Wolfram syndrome.
Nguyen LD, Fischer TT, Abreu D, Arroyo A, Urano F, Ehrlich BE. Nguyen LD, et al. Proc Natl Acad Sci U S A. 2020 Jul 21;117(29):17389-17398. doi: 10.1073/pnas.2007136117. Epub 2020 Jul 6. Proc Natl Acad Sci U S A. 2020. PMID: 32632005 Free PMC article.
Wolfram syndrome is a rare multisystem disease characterized by childhood-onset diabetes mellitus and progressive neurodegeneration. Most cases are attributed to pathogenic variants in a single gene, Wolfram syndrome 1 (WFS1). There currently is …
Wolfram syndrome is a rare multisystem disease characterized by childhood-onset diabetes mellitus and progressive neuro …
A mutant wfs1 zebrafish model of Wolfram syndrome manifesting visual dysfunction and developmental delay.
Cairns G, Burté F, Price R, O'Connor E, Toms M, Mishra R, Moosajee M, Pyle A, Sayer JA, Yu-Wai-Man P. Cairns G, et al. Sci Rep. 2021 Oct 14;11(1):20491. doi: 10.1038/s41598-021-99781-0. Sci Rep. 2021. PMID: 34650143 Free PMC article.
Wolfram syndrome (WS) is an ultra-rare progressive neurodegenerative disorder defined by early-onset diabetes mellitus and optic atrophy. ...There is limited availability of human ocular and brain tissues, and there are few animal models for WS that re …
Wolfram syndrome (WS) is an ultra-rare progressive neurodegenerative disorder defined by early-onset diabetes mellitus and opt …
29 results