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Open-label add-on treatment trial of minocycline in fragile X syndrome.
Open accessAbstract
BACKGROUND: Fragile X syndrome (FXS) is a disorder characterized by a variety of disabilities, including cognitive deficits, attention-deficit/hyperactivity disorder, autism, and other socio-emotional problems. It is hypothesized that the absence of the fragile X mental retardation protein (FMRP) leads to higher levels of matrix metallo-proteinase-9 activity (MMP-9) in the brain. Minocycline inhibits MMP-9 activity, and alleviates behavioural and synapse abnormalities in fmr1 knockout mice, an established model for FXS. This open-label add-on pilot trial was conducted to evaluate safety and efficacy of minocycline in treating behavioural abnormalities that occur in humans with FXS. METHODS: Twenty individuals with FXS, ages 13-32, were randomly assigned to receive 100 mg or 200 mg of minocycline daily. Behavioural evaluations were made prior to treatment (baseline) and again 8 weeks after daily minocycline treatment. The primary outcome measure was the Aberrant Behaviour Checklist-Community Edition (ABC-C) Irritability Subscale, and the secondary outcome measures were the other ABC-C subscales, clinical global improvement scale (CGI), and the visual analog scale for behaviour (VAS). Side effects were assessed using an adverse events checklist, a complete blood count (CBC), hepatic and renal function tests, and antinuclear antibody screen (ANA), done at baseline and at 8 weeks. RESULTS: The ABC-C Irritability Subscale scores showed significant improvement (p < 0.001), as did the VAS (p = 0.003) and the CGI (p < 0.001). The only significant treatment-related side effects were minor diarrhea (n = 3) and seroconversion to a positive ANA (n = 2). CONCLUSIONS: Results from this study demonstrate that minocycline provides significant functional benefits to FXS patients and that it is well-tolerated. These findings are consistent with the fmr1 knockout mouse model results, suggesting that minocycline modifies underlying neural defects that account for behavioural abnormalities. A placebo-controlled trial of minocycline in FXS is warranted. TRIAL REGISTRATION: ClinicalTrials.gov Open-Label Trial NCT00858689.
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“Reduction of MMP-9 levels with minocycline in humans with FXS (Dziembowska et al., 2013) contributes to reversal of several FXS phenotypes, including behavioral deficits and enhanced auditory electrocortical activity in adults and/or adolescents with FXS (Paribello et al., 2010; Leigh et al., 2013; Schneider et al., 2013).”
From The Perineuronal ‘Safety’ Net? Perineuronal Net Abnormalities in Neurological Disorders (2018) · cites this paper as Paribello et al., 2010
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“an open trial in adolescent and adult males with FXS demonstrated efficacy in all of the outcome measures (127).”
From Fragile X syndrome: causes, diagnosis, mechanisms, and therapeutics (2012) · cites this paper as [127]
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Metadata
- Authors
- Paribello, C., Tao, L., Folino, A., Berry-Kravis, E., Tranfaglia, M., Ethell, I. M., & Ethell, D. W.
- Affiliations
- Surrey Place Centre (CA)
- Fraxa Research Foundation (US)
- University of California, Riverside (US)
- Corresponding author
- Paribello, Carlo
- Venue
- BMC Neurology
- Published
- 2010-10-11
- Volume
- 10
- Issue
- 1
- Pages
- 91-91
- ISSN
- 1471-2377 (ISSN-L)
- PMC
- PMC2958860
- OpenAlex
- W2153053291
- Cited by
- 223
- References
- 45
- Citations by year
- 2026: 3 · 2025: 3 · 2024: 7 · 2023: 5 · 2022: 9 · 2021: 4 · 2020: 15 · 2019: 10 · 2018: 12 · 2017: 20 · 2016: 12 · 2015: 17 · 2014: 26 · 2013: 30 · 2012: 23
- FWCI
- 14.38
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- Locations
- BMC Neurology (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion)
- eScholarship (California Digital Library) (submittedVersion)
- Funders
- FRAXA Research Foundation
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Down syndrome and intellectual disability research
- Keywords
- fragile X syndrome, minocycline, MMP-9, FMRP, behavioural abnormalities, irritability, adjunctive therapy, drug safety, adverse events, antinuclear antibodies, Fmr1 knockout mice
- MeSH terms
- Adolescent, Adult, Enzyme Inhibitors, Female, Fragile X Syndrome, Humans, Male, Minocycline, Pilot Projects, Treatment Outcome, Young Adult
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