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Table 1 Characteristics of studies

From: Systematic review of pharmacological treatments in fragile X syndrome

 

Folic acid (FA)

  

Reference

Participants and Follow-Up Period

Methods

Interventions

Brown 1984 [21]

N = 2 brothers, 10.5 and 18.8 years. Setting: in-patients. FUP: 16 days

R, CO, DB. AASG: no; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

FA 1,6 mg/kg intravenous vs Placebo

Brown 1986 [22]

N = 5 males, 8 to 26 years. 3 autism FUP: 9 months

R, CO, DB. AASG: unclear; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

FA 250 mg/day vs Placebo

Carpenter 1983 [23]

N = 4 males, 2 to 10 years. IQs: 45 to 50. 2 autistic behaviour. FUP: 6 months

NR, CO, DB. AASG: no; AAC: unclear; AB: unclear; IDAA: unclear; FSR: unclear.

FA 10 mg/day vs Placebo

Fisch 1988 [24]

N = 6 males, 3.5 to 15 years. 3 autistic, 3 mental retardation only (borderline to severe). FUP: 12 months

R, CO, DB, SA. AASG: unclear; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

FA 10 mg/day vs Placebo

Froster-Iskenius 1986 [29]

N = 10 males, 15 to 54 years. IQ: 21 to 60. FUP: 4 months

R, P, DB. AASG: unclear; AAC: unclear; AB: no; IDAA: yes; FSR: yes.

FA 10 mg/day vs 0.0015 mg of folic acid/day, 2 months and 2 months with folic acid, 10 mg/day

Gillberg 1986 [30]

N = 4 males, 6 to 14 years. IQ: 39 to 64. All autism, 3 epilepsy. FUP: 9 months

NR, CO, DB, SA. AASG: no; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

FA 5 mg/day vs Placebo

Hagerman 1986 [25]

N = 25 males, 1 to 31 years. IQ: 39 to 82. Many of the younger ones in special education program with language and occupational therapy. Many adults use phenothiazines. FUP: 12 months

R, CO, DB, SA. AASG: yes; AAC: unclear; AB: yes; IDAA: yes; FSR: yes.

FA 10 mg/day vs Placebo

Madison 1986 [26]

N = 3 males, 3, 8 and 12 years old.

Moderate mental retardation. FUP: 11 weeks

NR, DB. AASG: no; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

No drug-no placebo (20 days). Placebo (13-45 days). Folic acid 10 mg/day (10-43 days)

Rosenblatt 1985 [28]

N = 2 monozygous male twins, 14 years old, mentally retarded. FU: 12 months

NR, CO. AASG: no; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

FA 5 mg/day vs Placebo

Strom 1992 [27]

N = 21 males, 2-22 years, mean 8.3 years. FUP: 24 weeks

R, CO, DB, SA. AASG: unclear; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

FA 15 mg/day vs Placebo

 

Ampakine compound CX516

  

Berry-Kravis 2006 [14]

N = 49. 11 female, 18-49 years. IQ: 36 to75, mean 43; 12 ≥50. Autism 21, spectrum: 4. 27 on psychoactive medication. FUP: 4 weeks

Phase II, R, P, DB, SA. AASG: yes; AAC: yes; AB: yes; IDAA: yes; FSR: yes.

Ampakine compound CX516 600-900 mg/day vs Placebo

 

Dextroamphetamine Methylphenidate

  

Hagerman 1988 [5]

N = 15, 2 female. 3.8-11.8 years. IQ: 29 to 77 (3 IQ >70). All attentional problems. 40% on stimulants. FUP: 3 weeks

R, CO, DB, SA. AASG: unclear; AAC: unclear; AB: yes; IDAA: yes; FSR: yes.

Dextroamphetamine

2 mg/kg/day vs Methylphenidate 0.6 mg/kg/day vs Placebo

 

L-Acetylcarnitine

  

Torrioli 1999 [32]

N = 20 males, 6-13 years. IQ:30 to 69

Hyperactive behaviour. FUP: 12 months

R, P, DB, SA. AASG: unclear; AAC: unclear; AB: unclear; IDAA: unclear; FSR: yes.

L-Acetylcarnitine 100 mg/kg/day vs Placebo

Torrioli 2008 [13]

N = 63 males, 6-13 years, all with Attention Deficit Hyperactivity Disorder. All extra care and stimulation at school and home, and visits by neuropsychiatrists. FUP: 12 months

Phase II, R, P, DB, SA. AASG: unclear; AAC: unclear; AB: unclear; IDAA: yes; FSR: yes.

L-Acetylcarnitine

20-50 mg/kg/day vs Placebo

  1. * AAC: adequate allocation concealment; AASG: adequate allocation sequence generation; AB: adequate blinding of participants, personnel and assessors; CO: crossover; DB: double blind; FSR: free of selective outcome reporting; FUP: follow up period; IDAA: incomplete data adequately addressed; N: number of patients; NR: non randomized; P: parallel; R: randomized; SA: statistical analysis presented.