Alcohol use dependence in fragile X syndrome

Salcedo-Arellano, María Jimena; Lozano, Reymundo; Tassone, Flora; Hagerman, Randi J.; Saldarriaga, Wilmar

doi:10.5582/irdr.2016.01046

Cited by 10 publications

(10 citation statements)

References 36 publications

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“…It might be for these negative effects that, in contrast to cases of high-functioning autism, where patients used drinking to cope with social anxiety (Lalanne et al, 2015 ), alcoholism is at least in FXS patients rare (cp. Salcedo-Arellano et al, 2016 ).…”

Section: The Fragile X Syndrome — Of Menmentioning

confidence: 99%

“…The relevance of the latter is further emphasized by the fact that alcohol abuse seemed to be involved in the case of severe FXTAS reported by Loesch and colleagues (Loesch et al, 2012 ). Recent studies revealed that alcohol is in fact exaggerating behavioral problems such as aggression and impulsivity in FXS patients (Salcedo-Arellano et al, 2016 ) and accelerating neurological deterioration in FXTAS (Muzar et al, 2014 ). It might be for these negative effects that, in contrast to cases of high-functioning autism, where patients used drinking to cope with social anxiety (Lalanne et al, 2015 ), alcoholism is at least in FXS patients rare (cp.…”

Section: The Fragile X Syndrome — Of Menmentioning

confidence: 99%

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Of Men and Mice: Modeling the Fragile X Syndrome

Dahlhaus

2018

Front. Mol. Neurosci.

109

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The Fragile X Syndrome (FXS) is one of the most common forms of inherited intellectual disability in all human societies. Caused by the transcriptional silencing of a single gene, the fragile x mental retardation gene FMR1, FXS is characterized by a variety of symptoms, which range from mental disabilities to autism and epilepsy. More than 20 years ago, a first animal model was described, the Fmr1 knock-out mouse. Several other models have been developed since then, including conditional knock-out mice, knock-out rats, a zebrafish and a drosophila model. Using these model systems, various targets for potential pharmaceutical treatments have been identified and many treatments have been shown to be efficient in preclinical studies. However, all attempts to turn these findings into a therapy for patients have failed thus far. In this review, I will discuss underlying difficulties and address potential alternatives for our future research.

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Section: The Fragile X Syndrome — Of Menmentioning

confidence: 99%

Section: The Fragile X Syndrome — Of Menmentioning

confidence: 99%

Of Men and Mice: Modeling the Fragile X Syndrome

Dahlhaus

2018

Front. Mol. Neurosci.

109

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“…A prospective open-label trial for 10 weeks in children ages 6 to 12 years with FXS showed treatment response in 9 of the 12 subjects (75%) in social behavior and inattention/hyperactivity using multiple standard behavior outcome measures; moreover, the drug was well tolerated with no severe or serious adverse effects reported [72]. Acamprosate is suggested to be the preferred targeted treatment for those with FXS and comorbid alcohol dependence [73]. This medication is also currently available clinically.…”

Section: Currently Available Targeted Treatments For Fragile X Syndromementioning

confidence: 99%

New Targeted Treatments for Fragile X Syndrome

Protić

Salcedo-Arellano

et al. 2019

CPR

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Fragile X Syndrome (FXS) is the most common cause of inherited intellectual disability with prevalence rates estimated to be 1:5,000 in males and 1:8,000 in females. The increase of >200 Cytosine Guanine Guanine (CGG) repeats in the 5’ untranslated region of the Fragile X Mental Retardation 1 (FMR1) gene results in transcriptional silencing on the FMR1 gene with a subsequent reduction or absence of fragile X mental retardation protein (FMRP), an RNA binding protein involved in the maturation and elimination of synapses. In addition to intellectual disability, common features of FXS are behavioral problems, autism, language deficits and atypical physical features. There are still no currently approved curative therapies for FXS, and clinical management continues to focus on symptomatic treatment of comorbid behaviors and psychiatric problems. Here we discuss several treatments that target the neurobiological pathway abnormal in FXS. These medications are clinically available at present and the data suggest that these medications can be helpful for those with FXS.

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“…Acamprosate has also been proposed as a beneficial treatment for patients with FXS syndrome diagnosed with alcohol dependency because it is helpful for both problems. 82…”

Section: Acamprosatementioning

confidence: 99%

Fragile X Syndrome and Targeted Treatments

Tassanakijpanich

Cabal-Herrera²,

Salcedo-Arellano

et al. 2020

J. Biomed. Transl. Res.

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Many targeted treatment studies have been carried out in individuals with Fragile X Syndrome (FXS) guided by animal studies from the Fragile X Mental Retardation 1 (FMR1) knock out (KO) mice and the fragile X Drosophila studies. Here we review the many medications that have been studied in patients with FXS and some of these medications are available for clinical use by wise clinicians. Other medications are not currently available by prescription because they are not approved by the FDA. No medication has received specific approval for treatment of FXS, although some have shown benefit from clinical studies. There is much to be done in the treatment of those with FXS and this report describes those pharmacological treatments that target the neurobiological mechanisms that are dysregulated by the lack of the Fragile X Protein (FMRP) in those with FXS.

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Alcohol use dependence in fragile X syndrome

Cited by 10 publications

References 36 publications

Of Men and Mice: Modeling the Fragile X Syndrome

Of Men and Mice: Modeling the Fragile X Syndrome

New Targeted Treatments for Fragile X Syndrome

Fragile X Syndrome and Targeted Treatments

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