Literature DB >> 22167274

Females experience a more severe disease course in Batten disease.

Jennifer Cialone1, Heather Adams, Erika F Augustine, Frederick J Marshall, Jennifer M Kwon, Nicole Newhouse, Amy Vierhile, Erika Levy, Leon S Dure, Katherine R Rose, Denia Ramirez-Montealegre, Elisabeth A de Blieck, Jonathan W Mink.   

Abstract

Juvenile neuronal ceroid lipofuscinosis (JNCL; CLN3 disease; Batten disease) is an autosomal recessive neurodegenerative disease of childhood. Symptoms typically present at school age with vision loss followed by progressive cognitive decline, motor dysfunction, seizures, and behavior problems. Studies on sex differences in JNCL have yielded mixed results, but parent anecdotes suggest that females experience a more precipitous disease course. Therefore, we sought to determine if sex-based differences exist in JNCL. We used data from the Unified Batten Disease Rating Scale (UBDRS), the Batten Disease Support and Research Association (BDSRA) database, and the PedsQL quality of life (QoL) survey to evaluate sex-based differences in functional independence and time from symptom onset to death. On average, females had JNCL symptom onset one year later and death one year earlier than did males. Despite a later age at onset, females had lower functional capability, earlier loss of independent function, and lower physical QoL. Future research in sex differences in JNCL may help to further understand the biological mechanisms underpinning the disease course and may point to targeted therapies.

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Year:  2011        PMID: 22167274      PMCID: PMC3320704          DOI: 10.1007/s10545-011-9421-6

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  19 in total

Review 1.  Estrogen actions in the central nervous system.

Authors:  B S McEwen; S E Alves
Journal:  Endocr Rev       Date:  1999-06       Impact factor: 19.871

2.  Homogeneous polymerase chain reaction nucleobase quenching assay to detect the 1-kbp deletion in CLN3 that causes Batten disease.

Authors:  Paul G Rothberg; Denia Ramirez-Montealegre; Sharon D Frazier; David A Pearce
Journal:  J Mol Diagn       Date:  2004-08       Impact factor: 5.568

3.  Distinct patterns of serum immunoreactivity as evidence for multiple brain-directed autoantibodies in juvenile neuronal ceroid lipofuscinosis.

Authors:  M J Lim; J Beake; E Bible; T M Curran; D Ramirez-Montealegre; D A Pearce; J D Cooper
Journal:  Neuropathol Appl Neurobiol       Date:  2006-10       Impact factor: 8.090

4.  Genotype does not predict severity of behavioural phenotype in juvenile neuronal ceroid lipofuscinosis (Batten disease).

Authors:  Heather R Adams; Christopher A Beck; Erika Levy; Rachel Jordan; Jennifer M Kwon; Frederick J Marshall; Amy Vierhile; Erika F Augustine; Elisabeth A de Blieck; David A Pearce; Jonathan W Mink
Journal:  Dev Med Child Neurol       Date:  2010-02-19       Impact factor: 5.449

Review 5.  Sex and systemic lupus erythematosus: the role of the sex hormones estrogen and prolactin on the regulation of autoreactive B cells.

Authors:  Christine M Grimaldi
Journal:  Curr Opin Rheumatol       Date:  2006-09       Impact factor: 5.006

6.  A clinical rating scale for Batten disease: reliable and relevant for clinical trials.

Authors:  F J Marshall; E A de Blieck; J W Mink; L Dure; H Adams; S Messing; P G Rothberg; E Levy; T McDonough; J DeYoung; M Wang; D Ramirez-Montealegre; J M Kwon; D A Pearce
Journal:  Neurology       Date:  2005-07-26       Impact factor: 9.910

Review 7.  Neurotrophic and neuroprotective actions of estrogen: basic mechanisms and clinical implications.

Authors:  Darrell W Brann; Krishnan Dhandapani; Chandramohan Wakade; Virendra B Mahesh; Mohammad M Khan
Journal:  Steroids       Date:  2007-02-21       Impact factor: 2.668

Review 8.  Estrogen actions in the brain and the basis for differential action in men and women: a case for sex-specific medicines.

Authors:  Glenda E Gillies; Simon McArthur
Journal:  Pharmacol Rev       Date:  2010-04-14       Impact factor: 25.468

9.  Neuropsychological symptoms of juvenile-onset batten disease: experiences from 2 studies.

Authors:  Heather R Adams; Jennifer Kwon; Frederick J Marshall; Elisabeth A de Blieck; David A Pearce; Jonathan W Mink
Journal:  J Child Neurol       Date:  2007-05       Impact factor: 1.987

Review 10.  Sexual dimorphism in autoimmune disease.

Authors:  P A McCombe; J M Greer; I R Mackay
Journal:  Curr Mol Med       Date:  2009-12       Impact factor: 2.222

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  32 in total

1.  Age-dependent therapeutic effect of memantine in a mouse model of juvenile Batten disease.

Authors:  Attila D Kovács; Angelika Saje; Andrew Wong; Serena Ramji; Jonathan D Cooper; David A Pearce
Journal:  Neuropharmacology       Date:  2012-06-06       Impact factor: 5.250

2.  Plasma biomarkers for neuronal ceroid lipofuscinosis.

Authors:  Samantha L Hersrud; Ryan D Geraets; Krystal L Weber; Chun-Hung Chan; David A Pearce
Journal:  FEBS J       Date:  2015-12-17       Impact factor: 5.542

3.  Genetic abnormalities and prognosis in patients with congenital and infantile nephrotic syndrome.

Authors:  Onur Cil; Nesrin Besbas; Ali Duzova; Rezan Topaloglu; Amira Peco-Antić; Emine Korkmaz; Fatih Ozaltin
Journal:  Pediatr Nephrol       Date:  2015-02-27       Impact factor: 3.714

Review 4.  Classification and natural history of the neuronal ceroid lipofuscinoses.

Authors:  Jonathan W Mink; Erika F Augustine; Heather R Adams; Frederick J Marshall; Jennifer M Kwon
Journal:  J Child Neurol       Date:  2013-07-09       Impact factor: 1.987

Review 5.  Neurobehavioral features and natural history of juvenile neuronal ceroid lipofuscinosis (Batten disease).

Authors:  Heather R Adams; Jonathan W Mink
Journal:  J Child Neurol       Date:  2013-09       Impact factor: 1.987

6.  Clinical trials in rare disease: challenges and opportunities.

Authors:  Erika F Augustine; Heather R Adams; Jonathan W Mink
Journal:  J Child Neurol       Date:  2013-09       Impact factor: 1.987

7.  Methodology of clinical research in rare diseases: development of a research program in juvenile neuronal ceroid lipofuscinosis (JNCL) via creation of a patient registry and collaboration with patient advocates.

Authors:  Elisabeth A de Blieck; Erika F Augustine; Frederick J Marshall; Heather Adams; Jennifer Cialone; Leon Dure; Jennifer M Kwon; Nicole Newhouse; Katherine Rose; Paul G Rothberg; Amy Vierhile; Jonathan W Mink
Journal:  Contemp Clin Trials       Date:  2013-04-26       Impact factor: 2.226

Review 8.  Sex differences in neurodevelopmental and neurodegenerative disorders: Focus on microglial function and neuroinflammation during development.

Authors:  Richa Hanamsagar; Staci D Bilbo
Journal:  J Steroid Biochem Mol Biol       Date:  2015-10-23       Impact factor: 4.292

9.  Short-Term Administration of Mycophenolate Is Well-Tolerated in CLN3 Disease (Juvenile Neuronal Ceroid Lipofuscinosis).

Authors:  Erika F Augustine; Christopher A Beck; Heather R Adams; Sara Defendorf; Amy Vierhile; Derek Timm; Jill M Weimer; Jonathan W Mink; Frederick J Marshall
Journal:  JIMD Rep       Date:  2018-06-20

Review 10.  Vision loss in juvenile neuronal ceroid lipofuscinosis (CLN3 disease).

Authors:  Madhu M Ouseph; Mark E Kleinman; Qing Jun Wang
Journal:  Ann N Y Acad Sci       Date:  2016-01-08       Impact factor: 5.691

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