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Evaluating the Neurophysiologic and Clinical Effects of Single Dose Drug Challenge

Phase 2
Recruiting
Conditions
Fragile X Syndrome
Interventions
Registration Number
NCT05418049
Lead Sponsor
Children's Hospital Medical Center, Cincinnati
Brief Summary

The aim of this study is to utilize neurophysiologic assessments, behavioral measures and clinical measures to assess how much deficits associated with Fragile X Syndrome from pre-dose to post-dose using pharmacology.

Detailed Description

Not available

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
45
Inclusion Criteria
  • Subjects ages 18-45, with FXS who completed the study entitled "Mechanisms and brain circuits underlying fragile X syndrome" (IRB # 2015-8425) or appropriate baseline measures through Biorepository (2013-7327).
  • FXS is defined as full FMR1 mutations (>200 CGG repeats) confirmed by genetic testing.
  • General good health as determined by physical exam, medical history and laboratory work up.
  • Stanford Binet IQ <85
  • Stable dosing of psychotropic drugs for at least 4 weeks.
Exclusion Criteria
  • Subjects with a history of intolerance to baclofen, roflumilast, or memantine will be excluded.
  • Subjects will also be excluded if they have taken any investigational drug within 3 months, have a history of substance abuse or dependence within 6 months, or significant psychiatric or CNS neurological disease unrelated to FXS.
  • Uncontrolled seizures or history of epilepsy with a seizure in the past 6 months
  • Auditory or visual impairments that cannot be corrected based on visual and auditory screener benchmarks.
  • Moderate to severe renal or hepatic impairment and determined by a study physician incorporating data from exam, medical history and laboratory value evaluation among other data points.
  • Use of barbiturates, benzodiazepines, antiepileptics, or other GABAergic or glutamatergic modulators
  • Current use of: Amifampridine, Butalbital, Codeine, Doxylamine, Ethanol, Hydrocodone, Isocarboxazid, Kava, Metoclopramide, Midazolam, Oxybate, Phenelzine, Promethazine, Thalidomide, Tranylcypromine, Trimethobenzamide, Erythromycin, Ketoconazole, Fluvoxamine, Enoxacin, and Cimetidine.
  • Those taking other psychiatric medications must be on stable doses for 4 weeks before the baseline visit.
  • Pregnancy or breast-feeding. For female subjects of child bearing potential, a urine pregnancy test will be performed.
  • Potential subjects with a creatinine clearance < 50 mL/min will be excluded.
  • Identified medical issues, inability to tolerate study procedures or study drug per the discretion of the Principal Investigator.

Study & Design

Study Type
INTERVENTIONAL
Study Design
CROSSOVER
Arm && Interventions
GroupInterventionDescription
Experimental Study ParticipantsRoflumilastParticipants received, in random order, a single dose of placebo, baclofen, roflumilast or memantine with a two-week washout period between doses.
Control Study ParticipantsPlaceboParticipants received, in random order, a single dose of placebo, baclofen, roflumilast or memantine with a two-week washout period between doses.
Experimental Study ParticipantsBaclofenParticipants received, in random order, a single dose of placebo, baclofen, roflumilast or memantine with a two-week washout period between doses.
Experimental Study ParticipantsMemantineParticipants received, in random order, a single dose of placebo, baclofen, roflumilast or memantine with a two-week washout period between doses.
Primary Outcome Measures
NameTimeMethod
Change in EEG Relative Gamma PowerPre-dose, 3-hour post-dose

EEG relative gamma power at rest was calculated as the percent of power in the gamma frequencies relative to the sum of power in all frequency bands, averaged across electrodes, and calculated separately at pre-dose and post-dose timepoints. To assess the impact of drug, the pre-dose relative gamma power was subtracted from post-dose relative gamma power. Higher numbers indicate more relative gamma power post-dose; lower numbers indicate more relative gamma power pre-dose.

Secondary Outcome Measures
NameTimeMethod
Clinical Global Impressions-ImprovementPre-dose, 3-hour post-dose

The Clinical Global Impressions - Improvement (CGI-I) requires the clinician to assess how much the patient's illness has changed relative to pre-dose, from 1 (very much improved) to 7 (very much worse).

Clinical Global Impressions-Improvement-CaregiverPre-dose, 3-hour post-dose

The CGI-I requires the caregiver to assess how much the patient's illness has changed relative to pre-dose, from 1 (very much improved) to 7 (very much worse).

Visual Analog Scale - CaregiverPre-dose, 3-hour post-dose

The parent or caregiver will be asked to assess how much anxiety the participant is expressing on a line between two endpoints (no anxiety to worsened anxiety).

Trial Locations

Locations (1)

Cincinnati Children's Hospital Medical Center

🇺🇸

Cincinnati, Ohio, United States

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