Literature DB >> 35047275

A Rare Cause of Recurrent Febrile Encephalopathy in a Child: The Expanding Spectrum of ATP1A3 Mutations.

Saja Tahir1, Nidheesh Chencheri2, Abdalla A Abdalla3,4, Mohamed O E Babiker2.   

Abstract

ATP1A3 mutations have been recognized in infants and children presenting with a diverse group of neurological phenotypes, including rapid-onset dystonia parkinsonism (RDP), alternating hemiplegia of childhood (AHC), and cerebellar ataxia, areflexia, pes cavus, optic atrophy, and sensorineural hearing loss (CAPOS) syndrome. A new phenotype of fever-induced paroxysmal muscle weakness and encephalopathy (FIPWE) in patients with ATP1A3 mutations at c.2267G>A p residue 756H has been described most recently in few cases. Here, we report an additional case with an ATP1A3 mutation at c.2267G>A p residue 756H presenting with fever-induced paroxysmal muscle weakness and encephalopathy. To the best of our knowledge, this is the first reported case from the Middle East. This 18-month-old boy presented with recurrent, reversible fever-induced episodes of seizures, central hypotonia, areflexia, and developmental regression. The mainstay management for patients with ATP1A3 related diseases is symptomatic treatment as there is no specific proposed treatment. Aggressive management of febrile illness may be helpful in alleviating the symptoms.
Copyright © 2021, Tahir et al.

Entities:  

Keywords:  atp1a3; episodic encephalopathy; fever; genetics; hypotonia

Year:  2021        PMID: 35047275      PMCID: PMC8759976          DOI: 10.7759/cureus.20438

Source DB:  PubMed          Journal:  Cureus        ISSN: 2168-8184


  7 in total

1.  Paroxysmal features responding to flunarizine in a child with rapid-onset dystonia-parkinsonism.

Authors:  Stefania Fornarino; Michela Stagnaro; Martina Rinelli; Danilo Tiziano; Margherita M Mancardi; Maria Traverso; Edvige Veneselli; Elisa De Grandis
Journal:  Neurology       Date:  2014-05-02       Impact factor: 9.910

Review 2.  Early Diagnosis of CAPOS Syndrome Before Acute-Onset Ataxia-Review of the Literature and a New Family.

Authors:  Anna Duat Rodriguez; Michaela Prochazkova; Saturnino Santos Santos; Oscar Rubio Cabezas; Veronica Cantarin Extremera; Luis Gonzalez-Gutierrez-Solana
Journal:  Pediatr Neurol       Date:  2017-01-25       Impact factor: 3.372

3.  Fever-Induced Paroxysmal Weakness and Encephalopathy, a New Phenotype of ATP1A3 Mutation.

Authors:  Sho T Yano; Kenneth Silver; Richard Young; Suzanne D DeBrosse; Roseànne S Ebel; Kathryn J Swoboda; Gyula Acsadi
Journal:  Pediatr Neurol       Date:  2017-04-29       Impact factor: 3.372

Review 4.  The Genetic Homogeneity of CAPOS Syndrome: Four New Patients With the c.2452G>A (p.Glu818Lys) Mutation in the ATP1A3 Gene.

Authors:  Roderick P P W M Maas; Jolanda H Schieving; Meyke Schouten; Erik-Jan Kamsteeg; Bart P C van de Warrenburg
Journal:  Pediatr Neurol       Date:  2016-03-17       Impact factor: 3.372

Review 5.  ATP1A3 mutations in infants: a new rapid-onset dystonia-Parkinsonism phenotype characterized by motor delay and ataxia.

Authors:  Allison Brashear; Jonathan W Mink; Deborah F Hill; Niki Boggs; W Vaughn McCall; Mark A Stacy; Beverly Snively; Laney S Light; Kathleen J Sweadner; Laurie J Ozelius; Leslie Morrison
Journal:  Dev Med Child Neurol       Date:  2012-08-28       Impact factor: 5.449

Review 6.  Distinct neurological disorders with ATP1A3 mutations.

Authors:  Erin L Heinzen; Alexis Arzimanoglou; Allison Brashear; Steven J Clapcote; Fiorella Gurrieri; David B Goldstein; Sigurður H Jóhannesson; Mohamad A Mikati; Brian Neville; Sophie Nicole; Laurie J Ozelius; Hanne Poulsen; Tsveta Schyns; Kathleen J Sweadner; Arn van den Maagdenberg; Bente Vilsen
Journal:  Lancet Neurol       Date:  2014-05       Impact factor: 44.182

7.  Research conference summary from the 2014 International Task Force on ATP1A3-Related Disorders.

Authors:  Hendrik Rosewich; Matthew T Sweney; Suzanne DeBrosse; Kevin Ess; Laurie Ozelius; Eva Andermann; Frederick Andermann; Gene Andrasco; Alice Belgrade; Allison Brashear; Sharon Ciccodicola; Lynn Egan; Alfred L George; Aga Lewelt; Joshua Magelby; Mario Merida; Tara Newcomb; Vicky Platt; Dominic Poncelin; Sandra Reyna; Masayuki Sasaki; Marcio Sotero de Menezes; Kathleen Sweadner; Louis Viollet; Mary Zupanc; Kenneth Silver; Kathryn Swoboda
Journal:  Neurol Genet       Date:  2017-03-02
  7 in total

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