Updated report on tools to measure outcomes of clinical trials in fragile X syndrome

Table 3 Outcome measures of problem behaviors: focus on disruptive behaviors domain in fragile X syndrome

Subdomain	Outcome measure	Shorter term	Longer term	Quality of tools
Irritability/aggression
	ABC-C_I	Yes	Yes	+++ [54, 55, 104–110]
	ABC-Cfx_I	Yes	Yes	+++ [9, 46, 103, 111–113]
Stereotype/self-injury/aggression	BPI-S	Yes		+ [115–117]
Variety of behaviors	FXS Rating Scale	Yes	Yes	+ [63, 114]
Inattention	Conners/ADHD-RS	Yes		++ [44, 64, 120, 122, 126]
Hyperactivity/impulsivity	Conners/ADHD-RS	Yes		++ [44, 64, 120, 122, 126]
Hyperactivity/impulsivity/inattention	CBCL_ADHD Scale	Yes		++ [130, 131]
Hyperactivity/impulsivity/inattention^a	SNAP-IV	Yes		++ [40, 113, 267]
Anxiety
Social withdrawal/anxiety	ABC-C_SW	Yes	Yes	+++ [9, 19, 28, 46, 103, 111–113]
Social anxiety
	PARS	Yes	Yes	++ [134, 138]
	ADAMS	Yes	Yes	++ [13, 17, 113, 132, 138]
	ADAMS Social Avoidance	Yes	Yes	++ [17, 139]
	CBCL Withdrawn	Yes	Yes	+ [18, 19, 135]
	CBCL’s DSM-Anxiety	Yes		+ [136, 137]
	SCARED	Yes		+ [10, 13]
Repulsive/compulsive behavior	RBS-R	Yes		+ [15, 179–184]
Social reciprocity/avoidance	SRS	Yes	Yes	+/++ [15, 46, 78–81, 112, 113]

Abbreviations: ABC-C Aberrant Behavior Checklist-Community Edition, ABC-C_I ABC-C, Irritability subscale, ABC-C _{FX_} I ABC-C_, FXS-specific, Irritability subscale, BPI Behavior Problems Inventory, ADHD-RS Attention-Deficit Hyperactivity Rating Scale, CBCL_ADHD Scale Child Behavior Checklist_ADHD subscale, SNAP-IV Swanson, Nolan and Pelham Questionnaire-4th ed. ABC-C_SW ABC-C, Lethargy/Social Withdrawal subscale, PARS Pediatric Anxiety Rating Scale, ADAMS Anxiety Depression and Mood Scale, CBCL Withdrawn CBCL Withdrawn Subscale, CBCL’s DSM-Anxiety CBCL’s Diagnostic and Statistical Manual-Anxiety Subscale, SCARED Screen For Child Anxiety Related Emotional Disorders, RBS-R Repetitive Behavior Scale-Revised, SRS Social Responsive Scale, shorter term deemed suitable for clinical trials lasting <12 months; longer term deemed suitable for clinical trials lasting >=12 months. Grading system: +++ strong, ++ moderate, + limited evidence, ? unknown/poor methodological quality, blank cell no evidence available
^aOppositional and defiant items are also included

ISSN: 1866-1955