Literature DB >> 16566884

Oromotor and communication findings in joubert syndrome: further evidence of multisystem apraxia.

Barbara A Braddock1, Janet E Farmer, Kathleen M Deidrick, Jana M Iverson, Bernard L Maria.   

Abstract

This study provides descriptive information in the areas of oromotor abilities and communication to better understand the spectrum of disability in individuals with Joubert syndrome. Participants included 21 individuals with the diagnosis of Joubert syndrome (mean age 10.45 years). Participants completed oromotor and receptive language measures. In addition, all of the participants' speech and gesture communication from a narrative task was coded and analyzed from videotape. Caregivers reported the participants' level of fine and gross motor function. The results show that individuals with Joubert syndrome exhibit a distinct oromotor pattern consistent with verbal and lingual apraxias. Despite significant motor skills deficits and oculomotor apraxia, persons with Joubert syndrome produced gestures when communicating, and those whose speech was less intelligible used a higher rate of gesture compared with those with greater verbal output. These findings suggest a new form of apraxia not previously described in the condition and are consistent with previous research that suggests that persons with Joubert syndrome typically do not exhibit classic symptoms of autism spectrum disorder.

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Year:  2006        PMID: 16566884     DOI: 10.1177/08830738060210020501

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  10 in total

Review 1.  Joubert Syndrome and related disorders.

Authors:  Francesco Brancati; Bruno Dallapiccola; Enza Maria Valente
Journal:  Orphanet J Rare Dis       Date:  2010-07-08       Impact factor: 4.123

2.  When is biopsy-proven TIN not simply TIN? Answers.

Authors:  Nicholas Ware; Neil J Sebire; W K Chong; Rajesh Krishnan; Stephen D Marks
Journal:  Pediatr Nephrol       Date:  2016-10-07       Impact factor: 3.714

Review 3.  Healthcare recommendations for Joubert syndrome.

Authors:  Ruxandra Bachmann-Gagescu; Jennifer C Dempsey; Sara Bulgheroni; Maida L Chen; Stefano D'Arrigo; Ian A Glass; Theo Heller; Elise Héon; Friedhelm Hildebrandt; Nirmal Joshi; Dana Knutzen; Hester Y Kroes; Stephen H Mack; Sara Nuovo; Melissa A Parisi; Joseph Snow; Angela C Summers; Jordan M Symons; Wadih M Zein; Eugen Boltshauser; John A Sayer; Meral Gunay-Aygun; Enza Maria Valente; Dan Doherty
Journal:  Am J Med Genet A       Date:  2019-11-11       Impact factor: 2.802

Review 4.  Joubert syndrome: insights into brain development, cilium biology, and complex disease.

Authors:  Dan Doherty
Journal:  Semin Pediatr Neurol       Date:  2009-09       Impact factor: 1.636

5.  Association of common variants in the Joubert syndrome gene (AHI1) with autism.

Authors:  Ana I Alvarez Retuerto; Rita M Cantor; Joseph G Gleeson; Anna Ustaszewska; Wendy S Schackwitz; Len A Pennacchio; Daniel H Geschwind
Journal:  Hum Mol Genet       Date:  2008-09-09       Impact factor: 6.150

Review 6.  Inherited cerebrorenal syndromes.

Authors:  Scott J Schurman; Steven J Scheinman
Journal:  Nat Rev Nephrol       Date:  2009-09       Impact factor: 28.314

Review 7.  Joubert syndrome: congenital cerebellar ataxia with the molar tooth.

Authors:  Marta Romani; Alessia Micalizzi; Enza Maria Valente
Journal:  Lancet Neurol       Date:  2013-07-17       Impact factor: 44.182

8.  MKS1 regulates ciliary INPP5E levels in Joubert syndrome.

Authors:  Gisela G Slaats; Christine R Isabella; Hester Y Kroes; Jennifer C Dempsey; Hendrik Gremmels; Glen R Monroe; Ian G Phelps; Karen J Duran; Jonathan Adkins; Sairam A Kumar; Dana M Knutzen; Nine V Knoers; Nancy J Mendelsohn; David Neubauer; Sotiria D Mastroyianni; Julie Vogt; Lisa Worgan; Natalya Karp; Sarah Bowdin; Ian A Glass; Melissa A Parisi; Edgar A Otto; Colin A Johnson; Friedhelm Hildebrandt; Gijs van Haaften; Rachel H Giles; Dan Doherty
Journal:  J Med Genet       Date:  2015-10-21       Impact factor: 6.318

9.  Joubert syndrome: Clinical and radiological characteristics of nine patients.

Authors:  Ahmed Farag Elhassanien; Hesham Abdel-Aziz Alghaiaty
Journal:  Ann Indian Acad Neurol       Date:  2013-04       Impact factor: 1.383

10.  Joubert Syndrome: Ophthalmological Findings in Correlation with Genotype and Hepatorenal Disease in 99 Patients Prospectively Evaluated at a Single Center.

Authors:  Brian P Brooks; Wadih M Zein; Amy H Thompson; Maryam Mokhtarzadeh; Daniel A Doherty; Melissa Parisi; Ian A Glass; May C Malicdan; Thierry Vilboux; Meghana Vemulapalli; James C Mullikin; William A Gahl; Meral Gunay-Aygun
Journal:  Ophthalmology       Date:  2018-07-25       Impact factor: 12.079

  10 in total

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