Literature DB >> 27372099

Clinical Phenotype of Adult Fragile X Gray Zone Allele Carriers: a Case Series.

Sarah M Debrey1, Maureen A Leehey2, Olga Klepitskaya2, Christopher M Filley3,4, Raj C Shah5, Benzi Kluger3, Elizabeth Berry-Kravis6,7, Elaine Spector8, Flora Tassone9,10, Deborah A Hall11.   

Abstract

Considerable research has focused on patients with trinucleotide (CGG) repeat expansions in the fragile X mental retardation 1 (FMR1) gene that fall within either the full mutation (>200 repeats) or premutation range (55-200 repeats). Recent interest in individuals with gray zone expansions (41-54 CGG repeats) has grown due to reported phenotypes that are similar to those observed in premutation carriers, including neurological, molecular, and cognitive signs. The purpose of this manuscript is to describe a series of adults with FMR1 alleles in the gray zone presenting with movement disorders or memory loss. Gray zone carriers ascertained in large FMR1 screening studies were identified and their clinical phenotypes studied. Thirty-one gray zone allele carriers were included, with mean age of symptom onset of 53 years in patients with movement disorders and 57 years in those with memory loss. Four patients were chosen for illustrative case reports and had the following diagnoses: early-onset Parkinson disease (PD), atypical parkinsonism, dementia, and atypical essential tremor. Some gray zone carriers presenting with parkinsonism had typical features, including bradykinesia, rigidity, and a positive response to dopaminergic medication. These patients had a higher prevalence of peripheral neuropathy and psychiatric complaints than would be expected. The patients seen in memory clinics had standard presentations of cognitive impairment with no apparent differences. Further studies are necessary to determine the associations between FMR1 expansions in the gray zone and various phenotypes of neurological dysfunction.

Entities:  

Keywords:  Ataxia; Fragile X-associated tremor/ataxia syndrome; Movement disorders; Parkinsonism; Trinucleotide repeat expansion

Mesh:

Substances:

Year:  2016        PMID: 27372099     DOI: 10.1007/s12311-016-0809-6

Source DB:  PubMed          Journal:  Cerebellum        ISSN: 1473-4222            Impact factor:   3.847


  63 in total

1.  Immune mediated disorders in women with a fragile X expansion and FXTAS.

Authors:  Isha Jalnapurkar; Nuva Rafika; Flora Tassone; Randi Hagerman
Journal:  Am J Med Genet A       Date:  2014-11-14       Impact factor: 2.802

2.  Abnormal elevation of FMR1 mRNA is associated with psychological symptoms in individuals with the fragile X premutation.

Authors:  David Hessl; Flora Tassone; Danuta Z Loesch; Elizabeth Berry-Kravis; Maureen A Leehey; Louise W Gane; Ingrid Barbato; Cathlin Rice; Emma Gould; Deborah A Hall; James Grigsby; Jacob A Wegelin; Susan Harris; Foster Lewin; Dahlia Weinberg; Paul J Hagerman; Randi J Hagerman
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2005-11-05       Impact factor: 3.568

3.  Impairment in the cognitive functioning of men with fragile X-associated tremor/ataxia syndrome (FXTAS).

Authors:  Jim Grigsby; Angela G Brega; Sébastien Jacquemont; Danuta Z Loesch; Maureen A Leehey; Glenn K Goodrich; Randi J Hagerman; Jennifer Epstein; Rebecca Wilson; Jennifer B Cogswell; Tristan Jardini; Flora Tassone; Paul J Hagerman
Journal:  J Neurol Sci       Date:  2006-06-15       Impact factor: 3.181

4.  A shortened version of the motor section of the Unified Huntington's Disease Rating Scale.

Authors:  S Siesling; A H Zwinderman; J P van Vugt; K Kieburtz; R A Roos
Journal:  Mov Disord       Date:  1997-03       Impact factor: 10.338

5.  A novel FMR1 PCR method for the routine detection of low abundance expanded alleles and full mutations in fragile X syndrome.

Authors:  Stela Filipovic-Sadic; Sachin Sah; Liangjing Chen; Julie Krosting; Edward Sekinger; Wenting Zhang; Paul J Hagerman; Timothy T Stenzel; Andrew G Hadd; Gary J Latham; Flora Tassone
Journal:  Clin Chem       Date:  2010-01-07       Impact factor: 8.327

6.  Linking the FMR1 alleles with small CGG expansions with neurodevelopmental disorders: preliminary data suggest an involvement of epigenetic mechanisms.

Authors:  Danuta Z Loesch; David E Godler; Mahmoud Khaniani; Emma Gould; Freya Gehling; Cheryl Dissanayake; Trent Burgess; Flora Tassone; Richard Huggins; Howard Slater; K H Andy Choo
Journal:  Am J Med Genet A       Date:  2009-10       Impact factor: 2.802

7.  The prevalence by staged severity of various types of diabetic neuropathy, retinopathy, and nephropathy in a population-based cohort: the Rochester Diabetic Neuropathy Study.

Authors:  P J Dyck; K M Kratz; J L Karnes; W J Litchy; R Klein; J M Pach; D M Wilson; P C O'Brien; L J Melton; F J Service
Journal:  Neurology       Date:  1993-04       Impact factor: 9.910

8.  The epidemiology of major depressive disorder: results from the National Comorbidity Survey Replication (NCS-R).

Authors:  Ronald C Kessler; Patricia Berglund; Olga Demler; Robert Jin; Doreen Koretz; Kathleen R Merikangas; A John Rush; Ellen E Walters; Philip S Wang
Journal:  JAMA       Date:  2003-06-18       Impact factor: 56.272

9.  Neurodevelopmental disabilities in children with intermediate and premutation range fragile X cytosine-guanine-guanine expansions.

Authors:  Meredith M Renda; Robert G Voigt; Dusica Babovic-Vuksanovic; W Edward Highsmith; Sherry S Vinson; Christine M Sadowski; Randi J Hagerman
Journal:  J Child Neurol       Date:  2012-12-23       Impact factor: 1.987

10.  FMR1 CGG allele size and prevalence ascertained through newborn screening in the United States.

Authors:  Flora Tassone; Ka Pou Iong; Tzu-Han Tong; Joyce Lo; Louise W Gane; Elizabeth Berry-Kravis; Danh Nguyen; Lisa Y Mu; Jennifer Laffin; Don B Bailey; Randi J Hagerman
Journal:  Genome Med       Date:  2012-12-21       Impact factor: 11.117

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  10 in total

1.  Evidence for the role of FMR1 gray zone alleles as a risk factor for parkinsonism in females.

Authors:  Danuta Z Loesch; Flora Tassone; George D Mellick; Malcolm Horne; Justin P Rubio; Minh Q Bui; David Francis; Elsdon Storey
Journal:  Mov Disord       Date:  2018-07       Impact factor: 10.338

2.  Working memory and arithmetic impairments in children with FMR1 premutation and gray zone alleles.

Authors:  Aline Aparecida Silva Martins; Giulia Moreira Paiva; Carolina Guimarães Ramos Matosinho; Elisângela Monteiro Coser; Pablo Augusto de Souza Fonseca; Vitor Geraldi Haase; Maria Raquel Santos Carvalho
Journal:  Dement Neuropsychol       Date:  2022 Jan-Mar

3.  Child Challenging Behavior Influences Maternal Mental Health and Relationship Quality Over Time in Fragile X Syndrome.

Authors:  Heather Fielding-Gebhardt; Steven F Warren; Nancy C Brady
Journal:  J Autism Dev Disord       Date:  2020-03

4.  A methylation PCR method determines FMR1 activation ratios and differentiates premutation allele mosaicism in carrier siblings.

Authors:  Andrew G Hadd; Stela Filipovic-Sadic; Lili Zhou; Arianna Williams; Gary J Latham; Elizabeth Berry-Kravis; Deborah A Hall
Journal:  Clin Epigenetics       Date:  2016-12-01       Impact factor: 6.551

Review 5.  Fragile X syndrome: a review of clinical and molecular diagnoses.

Authors:  Claudia Ciaccio; Laura Fontana; Donatella Milani; Silvia Tabano; Monica Miozzo; Susanna Esposito
Journal:  Ital J Pediatr       Date:  2017-04-19       Impact factor: 2.638

6.  Curvilinear Association Between Language Disfluency and FMR1 CGG Repeat Size Across the Normal, Intermediate, and Premutation Range.

Authors:  Jessica Klusek; Anna Porter; Leonard Abbeduto; Tatyana Adayev; Flora Tassone; Marsha R Mailick; Anne Glicksman; Bridgette L Tonnsen; Jane E Roberts
Journal:  Front Genet       Date:  2018-08-24       Impact factor: 4.599

7.  Screening for FMR1 CGG Repeat Expansion in Thai Patients with Autism Spectrum Disorder.

Authors:  Areerat Hnoonual; Charunee Jankittunpaiboon; Pornprot Limprasert
Journal:  Biomed Res Int       Date:  2021-12-08       Impact factor: 3.411

8.  Maternal Mental Health and Parenting Stress and Their Relationships to Characteristics of the Child With Fragile X Syndrome.

Authors:  Lauren Bullard; Danielle Harvey; Leonard Abbeduto
Journal:  Front Psychiatry       Date:  2021-11-24       Impact factor: 4.157

9.  Connective Tissue Disorders and Fragile X Molecular Status in Females: A Case Series and Review.

Authors:  Merlin G Butler; Waheeda A Hossain; Jacob Steinle; Harry Gao; Eleina Cox; Yuxin Niu; May Quach; Olivia J Veatch
Journal:  Int J Mol Sci       Date:  2022-08-13       Impact factor: 6.208

10.  The Spectrum of Neurological and White Matter Changes and Premutation Status Categories of Older Male Carriers of the FMR1 Alleles Are Linked to Genetic (CGG and FMR1 mRNA) and Cellular Stress (AMPK) Markers.

Authors:  Danuta Z Loesch; Nicholas Trost; Minh Q Bui; Eleanor Hammersley; Sui T Lay; Sarah J Annesley; Oana Sanislav; Claire Y Allan; Flora Tassone; Zhi-Ping Chen; Kevin R W Ngoei; Bruce E Kemp; David Francis; Paul R Fisher; Elsdon Storey
Journal:  Front Genet       Date:  2018-11-12       Impact factor: 4.599

  10 in total

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