ARV1 mutation is known to present as developmental and epileptic encephalopathy (DEE)-38. However, the phenotypic spectrum has been expanding ever since it was reported in 2016. Along with seizures and developmental delay, other unique clinical features include ophthalmological abnormalities and movement disorders in the form of ataxia and dystonia, especially in those with missense mutation. These manifestations closely mimic ataxia telangiectasia. Elevation of alpha-fetoprotein levels is an important investigative marker in the diagnosis of ataxia telangiectasia and ataxia with oculomotor apraxia syndromes. ARV1 can also be associated with increased alpha-fetoprotein. There are no reports evaluating alpha-fetoprotein levels in cases with ARV1 mutation, which is significant in the context of ocular abnormalities with ataxia. We report a case of ARV1 mutation presenting with ataxia, ocular abnormalities, and elevated alpha-fetoprotein levels, thus mimicking autosomal recessive cerebellar ataxias. This study provides a comprehensive literature review of the cases reported so far, thus expanding the understanding of the spectrum of presentation, and helps in correlating the clinical picture with the underlying causative genetic mutation. ARV1 gene is another example of one gene with phenotypic pleiotropy. Though presentation with DEE is common, a few, especially those with missense mutations, can present with ataxia and ocular abnormalities. All cases presenting with ataxia who have increased alpha-fetoprotein levels and seizures should be tested for the ARV1 gene, when testing for ataxia genes is negative. The underlying genetic mechanism can explain the varying clinical manifestations of the ARV1 gene.
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http://dx.doi.org/10.1007/s12311-023-01606-5 | DOI Listing |
J Int Med Res
September 2024
Department of Respiratory and Critical Care Medicine, Yuyao People's Hospital of Zhejiang Province (Affiliated Yangming Hospital), Yuyao, China.
Objective: To investigate the signature genes of fatty acid metabolism and their association with immune cells in pulmonary arterial hypertension (PAH).
Methods: Fatty acid metabolism-related genes were obtained from the GeneCards database. In this retrospective study, a PAH-related dataset was downloaded from the Gene Expression Omnibus database and analyzed to identify differentially expressed genes (DEGs).
Cerebellum
June 2024
Department of Pediatric Neurology, Jawaharlal Nehru Medical College, KLE Academy of Higher Education and Research, Room No. 25, KLE's PK Hospital, Belagavi, Karnataka, 590010, India.
ARV1 mutation is known to present as developmental and epileptic encephalopathy (DEE)-38. However, the phenotypic spectrum has been expanding ever since it was reported in 2016. Along with seizures and developmental delay, other unique clinical features include ophthalmological abnormalities and movement disorders in the form of ataxia and dystonia, especially in those with missense mutation.
View Article and Find Full Text PDFEquine Vet J
May 2024
Department of Animal Molecular Biology, National Research Institute of Animal Production, Balice, Poland.
J Pers Med
March 2023
Central Laboratory, Guangxi Health Commission Key Laboratory of Glucose and Lipid Metabolism Disorders, The Second Affiliated Hospital of Guilin Medical University, Guilin 541000, China.
Background: is an herbal medicine with anti-cancer effects. However, little is known about the anti-cancer effect of on colorectal cancer. Therefore, our study aimed to investigate the specific molecular mechanism of inhibition of colorectal cancer.
View Article and Find Full Text PDFAppl Environ Microbiol
April 2022
State Key Laboratory of Food Science and Technology, Jiangnan University, Wuxi, China.
Saccharomyces cerevisiae is a well-performing workhorse in chemical production, which encounters complex environmental stresses during industrial processes. We constructed a multiple stress tolerance mutant, Med15, that was obtained by engineering the KIX domain of Mediator tail subunit Med15. Med15 interacted with transcription factor Hap5 to improve expression for sterol homeostasis for decreasing membrane fluidity and thereby enhancing acid tolerance.
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