Psychometric Properties of Lower Limb Somatosensory Function and Body Awareness Outcome Measures in Children with Upper Motor Neuron Lesions: A Systematic Review

Figures & data

Table 1. Studies characteristics and ICF-Code

Study references (author, year)	Study aim	Outcome Measure	Modality	Study population		ICF-Code
				Diagnoses, number	Age (y)
Uzun Akkaya et al.^Citation40	Investigate differences in somatosensory function in children with and without CP, and whether somatosensory impairments affect gait quality	6-item Semmes-Weinstein Monofilament test kit (2.83–6.65) Two-Point discrimination with a discriminator 128 Hz tuning fork Knee position: Active reproduction of 30° knee flexion position, while sitting without vision, measured with a digital goniometer	Tactile function Vibration Joint position sense	Children with unilateral CP, n = 15 Children with bilateral CP, n = 15 Typically developing children, n = 15	5–18	B265 B270 B260
Abdin et al.^Citation41	Assess correlations of the proprioception abilities from the more and less affected leg with postural balance	Knee position: Active reproduction of 45° knee flexion position, measured with a Biodex System 3 Pro isokinetic dynamometer	Joint position sense	Children with unilateral CP, n = 29 (GMFCS I, II)	6–14	B260
Zarkou et al.^Citation42	Assess correlations of somatosensory function, and walking abilities, and balance	6-item Semmes-Weinstein Monofilament test kit (2.83–6.65) Two-Point discrimination with a discriminator 128 Hz tuning fork Knee position: Active reproduction of 30° knee flexion position, while sitting without vision, measured with a digital goniometer	Tactile function Vibration Joint position sense	Children with bilateral CP, n = 10	9–19	B265 B270 B260
De Bode et al.^Citation43	Assessment of light touch and proprioception before and after ten days of intensive mobility training in individuals after hemispherectomy	Fugl-Meyer assessment Sensory scale Applying tactile stimulus to the thigh and foot sole. Testing the hip, knee, ankle joint, and the big toe in flexion, and extension.	Tactile function Joint movement sense (Kinesthesia)	Children after a cerebral hemispherectomy, n = 18	Unclear	B265 B260
Wingert et al.^Citation7	Assessing joint-position sense and kinesthesia in all extremities in participants with diplegic or hemiplegic CP	Hip position: Pointing task to target angles along the semi-goniometer axis, with and without vision of the leg Hip movement: Awareness of the passive moved limb direction, without vision of the leg	Joint position sense(Statesthesia) Joint movement sense (Kinesthesia)	Spastic bilateral CP (GMFCS I, II), n = 21 Unilateral CP; (GMFCS I, II), n = 17	7–35 8–27	B260 B260
McLaughlin et al.^Citation8	Assess lower extremity sensory function, and its feasibility, difference between children with and without CP, ability to measure change after a dorsal rhizotomy	Light touch: Stroking the child with a cotton applicator (mid-thigh L2-L3; mid-shin L3-4; mid dorsum of the foot L4-S1) Direction of scratch: Touch application of 2 cm, 5 cm, 10 cm distance with a wooden stick (mid-thigh L2-L3; mid-shin L3-4; mid dorsum of the foot L4-S1) Vibration sense: 128 Hz fork to the medial malleolus, tibial tuberosity, and first metatarsal head Knee position: Reproduce active knee angle of 15°,30°,45° knee extension, after passive elevation to these positions Toe movement: Identify movement of the toe up or down	Tactile function Vibration Tactile function Joint position sense Joint movement sense	Children with CP, n = 62 (Spastic bilateral, n = 48; unilateral, n = 10, athetoid, n = 3 hypotonia = 1) Typically developing children, n = 65	3–18	B265 B270 B260 B260
Chafetz et al.^Citation44	To assess within-rater agreement for the total scores of light touch and pinprick in children and youth	ISNCSCI sensory	Tactile function	Children and adolescents with spinal cord injury (n = 187)	15–19	B265
Mulcahey et al.^Citation45	To assess rater agreement for the ISNCSCI motor and sensory scores	ISNCSCI sensory	Tactile function	Adolescents with spinal cord injury (n = 5)	15–19	B265
Mulcahey et al.^Citation28	To assess the intra-rater reliability of the ISNCSCI in different age groups	ISNCSCI sensory	Tactile function	Children with spinal cord injury (n = 74)	4–21	B265
Nuara et al.^Citation46	Assessment of asymmetry in self-portraits in children with CP and typically developing peers	Self-portrait: Children made three drawings: a self-portrait, a portrait of their best classmate-fiend, and a portrait of a hemiparetic peer undergoing rehabilitation	Spatial body awareness	Unilateral CP, n = 10 Typically developing children, n = 17	5–11	B180
Asano et al.^Citation47	Assessing tactile localization task, as part of body image in a group of children with different levels of motor function, and investigate the relationship with gross motor function	Tactile localization task: Children had to point on a screen the body parts where the tester applied a tactile input	Spatial body awareness	Children with CP, n = 10, Children with autism spectrum disorder, n = 5 Children with intellectual disabilities, n = 3 Child with ADHD, n = 1 (GMFCS I–III)	3–12	B180
Fontes et al.^Citation9	Developing a battery of neuropsychological tasks that assess body schema, body image, and body structural description in children with and without CP	Visual body-parts localization task: The child has to recognize a body part in a picture and show it on his/her body Verbal body-parts localization task: The child has to show a part of his/her body that has been named to him/her Matching body parts by localization task: The child should show an adjacent body part from a presented body part on a screen. Matching body parts by function: The child arranges on presented body parts pictures on a screen with the same functions to each other Body parts and object association task: The child assigns depicted body parts to an associated task, presented on a screen Naming body parts. The child names body parts which are shown in pictures on a screen.	Spatial body awareness Structural body awareness	Unilateral CP (analysis of items), n = 12 Unilateral CP (psychometric analysis), n = 49 Typically developing children (feasibility), n = 30	5–10 5-13 4–6	B180

Abbreviations: ICF – International Classification of Functioning, Disability, and Health; ISNCSCI – International Standard for Neurological Classification of Spinal Cord Injury; ADHS – Attention Deficit Hyperactivity Disorder

Table 2. Results of studies on measurement properties, Risk of Bias, and methodological quality rating

M	Outcome Measure	Internal consistency			Reliability			Hypotheses testing			Responsiveness
		Qua-lity^a	Result	Rating (RoB)^b	Qua-lity^a	Result	Rating (RoB)^b	Qua-lity^a	Result	Rating (RoB)^b	Qua-lity^a	Result	Rating (RoB)^b
Tactile function	Monofilament^Citation40	Na	Na	Na	Na	Na	Na	?	rs 0.45* Differentiation between groups**	Doubt-ful	Na	Na	Na
	Monofilament^Citation42	Na	Na	Na	NA	Na	Na	?	BESTest overall: r = 0.00 ns COPA: rs 0.31 ns COPV: rs 0.01 ns	Doubt-ful	Na	Na	Na
	Two-point discrimination^Citation40	Na	Na	Na	Na	Na	Na	?	Differentiation between groups rs 0.30**	Doubt-ful	Na	Na	Na
	Two-point discrimination^Citation42	Na	Na	Na	Na	Na	Na	?	BESTest overall: rs 0.64*, COPA rs 0.86 * COPV: r 0.045	Doubt-ful	Na	Na	Na
	Fugl-Meyer assessment sensory scale^Citation43	Na	Na	NA	Na	Na	Na	Na	Na	Na	?	Before MT: 6.2 ± 1.9. After MT: 6.7 ± 1.6	Inade-quate
	Light touch^Citation8	Na	Na	Na	Na	Na	Na	-	Differentiation between groups ns	Doubt-ful	Na	Na	Na
	Direction of scratch^Citation8	Na	Na	Na	Na	Na	Na	+	Differentiation between groups**	Doubt-ful	Na	Na	Na
	ISNCSCI sensory^Citation44	Na	Na	Na	+	Tetraplegia: LT: ICC 0.97 (0.95–0.98) PP: ICC 0.95 (0.92–0.96) Paraplegia: LT: ICC 0.98 (0.97–0.99) PP: ICC 0.98 (0.96–0.98)	Inade-quate	Na	Na	Na	Na	Na	Na
	ISNCSCI sensory^Citation45	Na	Na	Na	+	Before Training: ICC 0.82 (0.56–0.98) After Training: ICC 0.77 (0.47–0.98)	Inade-quate	Na	Na	Na	Na	Na	Na
	ISNCSCI sensory^Citation28	Na	Na	Na	+	Tetraplegia: LT: ICC 0.98 (0.97–0.99) PP: ICC 0.98 (0.97–0.99) Paraplegia: LT: ICC 0.94 (0.90–0.97) PP: ICC 0.94 (0.89–0.97)	Inade-quate	Na	Na	Na	Na	Na	Na
Vibration	Vibration sense^Citation40	Na	Na	Na	Na	Na	Na	?	rs −0.35 ns Differentiation between groups ns	Doubt-ful	Na	Na	Na
	Vibration sense^Citation42	Na	Na	Na	Na	Na	Na	?	BESTest overall: rs −0.31 ns, COPA: rs 0.73**, COPV: rs 0.69*	Doubt-ful	Na	Na	Na
	Vibration sense^Citation8	Na	Na	Na	Na	Na	Na	?	Differentiation between groups*	Doubt-ful	Na	Na	Na
Joint position sense	Hip position^Citation7	Na	Na	Na	Na	Na	Na	+	With vision: CD-DD ns, CN-DN ns, CD-HD ns, CN-HN ns Without vision: CD-DD ns, CN-DN **, CD-HD **, CN-HN **	Doubt-ful	Na	Na	Na
	Knee position^Citation40	Na	Na	Na	Na	Na	Na	+	EVGS: rs 0.16 ns Differentiation between groups**	Doubt-ful	Na	Na	Na
	Knee position^Citation42	Na	Na	Na	Na	Na	Na	+	BESTest overall: rs −0.28; COPA: rs 0.23 COPV: rs −0.09	Doubt-ful	Na	Na	Na
	Knee position sense^Citation8	Na	Na	Na	Na	Na	Na	?	Differentiation between groups ns	Doubt-ful	Na	Na	Na
Joint movement sense	Fugl-Meyer assessment sensory scale^Citation43	Na	Na	NA	Na	Na	Na	Na	Na	Na	?	Before MT: 10.1 ± 3.4 After MT: 10.4 ± 3.9	Inade-quate
	Toe movement^Citation8	Na	Na	Na	Na	Na	Na	+	Differentiation between groups*	Doubt-ful	Na	Na	Na
	Hip movement^Citation7	Na	Na	Na	Na	Na	Na	+	CD-DD ns, CN-DN ns, CD-HD ns, CN-HN **	Doubt-ful	Na	Na	Na
Spatial body representation	Visual body parts localization^Citation48	?	0.67 K-R20	Very good	Na	Na	Na	+	Verbal body parts: localization r 31** Matching body parts by localization r 33**	Doubt-ful	Na	Na	Na
	Verbal body parts localization^Citation48	?	0.65 K-R20	Very good	Na	Na	Na	+	Visual body parts localization: r 27** Matching body parts by localization: r 33**	Doubt-ful	Na	Na	Na
	Matching body parts by localization^Citation48	?	0.60 K-R20	Very good	Na	Na	Na	+	Visual body parts localization: r 31** Verbal body parts localization: r 33**	Doubt-ful	Na	Na	Na
	Tactile localization task^Citation47	Na	Na	Na	Na	Na	Na	?	GMFM: 3-toe: r 0.24 ns, 5-toe: r 0.04 ns, LE: r 0.80** Differentiate between groups: toe ns; LE **	Doubt-ful	Na	Na	Na
	Self-portrait^Citation48	Na	Na	Na	Na	Na	Na	+	Differentiation between groups*	Very good	Na	Na	Na
Structural body representation	Matching body parts by function^Citation48	+	0.60 K-R20	Very good	Na	Na	Na	+	Naming body parts: r 0.21*	Doubt-ful	Na	Na	Na
	Body parts and object association task^Citation48	+	0.60 K-R20	Very good	Na	Na	Na	+	Naming body parts: r 0.21*	Doubt-ful	Na	Na	Na
	Naming body parts^Citation48	+	0.63 K-R20	Very good	Na	Na	Na	+	Body parts and object association: r 0.21*	Doubt-ful	Na	Na	Na

^amethodological quality rating of the result by COSMIN; sufficient (+); insufficient (-); or indeterminate (?)

^bCOSMIN Risk of Bias (RoB) checklist rating as for each item as very good; adequate; doubtful; inadequate; not applicable. Overall rating, as the worst score counts

Level of significance: *p < 0.01; **p < 0.001, ns- not significant

Abbreviations: M – Modality; Na – not applicable; r – Pearson correlation; r_s – Spearman correlation; ICC- Intraclass correlation coefficient; AI – Asymmetry Index; K-R20 – Kruder Richardson; MT – Mobility Therapy; BR – Body Representation; CD – Control Dominant side; DD – Diplegia Dominant side; CN – Control Nondominant side; DN – Diplegia Nondominant side; EVGS – Edinburgh Visual Gait Score; HD – Hemiplegia Dominant side; HN – Hemiplegia Nondominant side; BESTest – Balance Evaluation System Test, COPA – Center Of Pressure Area; COPV – Center Of Pressure Velocity; LT – Light touch; PP – Pin Prick; LE – Lower Extremity; GMFM – Gross Motor Function Measurement; ISNCSCI – International Standard for Neurological Classification of Spinal Cord Injury

Table 3. Best evidence Synthesis

Modality; OM	Quality of evidence^a	Internal consistency			Reliability			Hypotheses testing (Construct validity)			Responsiveness
		n	RoB	Quality/ Diagnosis	n	RoB	Quality/ Diagnosis	n	RoB	Quality/Diagnosis	n	RoB	Quality/Diagnosis
Tactile function: Monofilament^Citation40,Citation42	Very low	-	-	-	-	-	-	40	Doubtful	?/CP	-	-	-
Tactile function: Two-point discrimination^Citation40,Citation42	Very low	-	-	-	-	-	-	40	Doubtful	?/CP	-	-	-
Tactile function: Light touch, direction of scratch^Citation8,Citation43	Very low	-	-	-	-	-	-	62	Doubtful	?/CP	18	Inadequate	?/HS
Tactile function: ISNCSCI sensory^Citation28,Citation44,Citation45	Low	-	-	-	266	Inadequate	+/SCI	-	-	-	-	-	-
Vibration^Citation8,Citation40,Citation42	Very low	-	-	-	-	-	-	102	Doubtful	?/CP	-	-	-
JPS: Knee and Hip position^Citation7,Citation40–42	Low	-	-	-	-	-	-	140	Doubtful	+/CP	-	-	-
JMS: Toe and Hip movement^Citation7,Citation8,Citation43	Very low	-	-	-	-	-	-	100	Doubtful	?/CP	18	Inadequate	?/HS
Spatial body represenation^Citation48	Moderate	61	Very good	+/CP	-	-	-	61	Doubtful	+/CP	-	-	-
Structural body representation^Citation46–48	Moderate	61	Very good	+/CP	-	-	-	89	Doubtful	+/CP, ADHD, ASD, ID	-	-	-

^aThese definitions were adapted from the GRADE approach³⁸:

High = We are very confident that the true measurement property lies close to that of the estimate of the measurement property

Moderate = We are moderately confident in the measurement property estimate: the true measurement property is likely to be close to the estimate of the measurement property, but there is a possibility that it is substantially different; Low = Our confidence in the measurement property estimate is limited: the true measurement property may be substantially different from the estimate of the measurement property; Very low = We have very little confidence in the measurement property estimate: the true measurement property is likely to be substantially different from the estimate of the measurement property

Abbreviations: CP- Cerebral Palsy; HS – cerebral Hemispherectomies; OM – Outcome Measure; ISNCSCI – International Standard for Neurological Classification of Spinal Cord Injury; JPS – Joint Position Sense; JMS – Joint Movement Sense; ADHD – Attention deficit hyperactivity disorder; ASD – Autism Spectrum Disorder; ID – Intellectual Disabilities

Table 4. Information to extract on feasibility according to the COSMIN guidelines

Modality	Outcome Measure	Patient’s comprehensibility	Clinician’s comprehensibility	Type and ease of administration	Duration of the test^a	Evaluation of the test^b	Patient’s required mental and physical ability level	Ease of standardization	Copyright	Requirement and Cost of instrument
Tactile function	Monofilament^Citation40,Citation42	All children completed the test	Not stated	Not stated	Not stated	Not stated	Not stated	Not stated	No	Mono-filament set Around $50
	Two-pointdiscrimination^Citation40,Citation42	All children completed the test	Not stated	Not stated	Not stated	Not stated	Not stated	Not stated	No	Aesthesiometer, or discriminator Around $50
	Fugl-Meyer assessment sensory scale^Citation43	All children completed the test	No training	Manual test Easy	Not stated	Not stated	Sitting or lying position	Easy	No	Cotton applicator Test manual Around $2
	Light touch^Citation8	87% of the children with CP, and 95% TD children could successfully complete	No training	Manual test Easy	Not stated	Not stated	Minimum cognitive ability of a 5-year-old child Sitting position has to be possible	Easy	No	Cotton applicator Around $2
	Direction of scratch^Citation8	33% of the children with CP and 75% TD children could successfully complete	No training	Manual test Easy	Not stated	Not stated	Minimum cognitive ability of a 5-year-old child Sitting position has to be possible	Easy	No	Cotton applicator Around $2
	ISNCSCI sensory^Citation28,Citation44,Citation45	54% of children younger than 4y could be tested. High reliability values for children aged 6 years and older.	Special training recommender	Manual test Easy	Not stated	Not stated	Minimum cognitive ability of a 6-year-old child Lying position	Easy	No	Cotton applicator Around $2
Vibration	Vibration sense^Citation8,Citation40,Citation42	67% of the children with CP, and 89% TD children could successfully complete	No training	Manual test Easy	Not stated	Not stated	Minimum cognitive ability of a 5-year-old child Sitting position has to be possible	Easy	No	Vibration fork Around $50
Joint movement sense	Hip position^Citation7	All children, and young people could perform the test	Not stated	Mechanical test Not clear	Not stated	Not stated	Sitting position	Not clear	Not stated	Custom built device Costs not available
	Knee position^Citation40,Citation42	All children could perform the test	Not stated	Mechanical test Not clear	Not stated	Not stated	Sitting position	Not clear	Not stated	Electro-goniometer Around $25
	Knee position^Citation41	All children could perform the test	Not stated	Mechanical test Not clear	Not stated	Not stated	Sitting position	Not clear	Not stated	Biodex system 3 Pro Isokinetic dynamo-meter $5000-70000
	Knee position^Citation8	68% of the children with CP, and 79% TD children could complete	No training	Manual test Easy	Not stated	Not stated	Minimum cognitive ability of a 5-year-old child Sitting position has to be possible	Easy	No	No equipment
Joint movement sense	Fugl-Meyer assessment sensory scale^Citation43	All children could perform the test	No training	Manual test Easy	Not stated	Not stated	Supine lying position	Easy	No	No cost
	Toe movement^Citation8	78% of the children with CP, and 95% TD children could complete	No training	Manual test Easy	Not stated	Not stated	Minimum cognitive ability of a 5-year-old child Sitting position has to be possible	Easy	No	No equipment
	Hip movement^Citation7	All children, and young people could perform the test	Not stated	Mechanical test Not clear	Not stated	Not stated	Sitting position	Not clear	Not stated	Custom built device
Spatial body representation	Self-portrait^Citation46	53% of the children with CP, and 89% TD children could complete	No training	Self-portrait Easy	Not stated	Not stated	Sitting position	Easy	No	Pencil, and sheet
	Tactile localization task^Citation47	All children completed the test	Not clear	Computer program Not clear	Not stated	Not stated	Sitting position	Not clear	Program notavailable	Computer program
	Visual body parts localization^Citation48	All children completed the test	Not clear	Computer program Not clear	Not stated	Not stated	Sitting position	Not clear	Program not available	Computer, program
	Verbal body parts localization^Citation48	All children completed the test	Not clear	Not clear	Not stated	Not stated	Sitting position	Not clear	Program not available	-
	Matching body parts by localization^Citation48	All children completed the test	Not clear	Computer program Not clear	Not stated	Not stated	Sitting position	Not clear	Program not available	Computer, program
Structural body representation	Matching body parts by function^Citation48	All children completed the test	Not clear	Computer program Not clear	Not stated	Not stated	Sitting position	Not clear	Program not available	Computer, program
	Body parts and object association task^Citation48	All children completed the test	Not clear	Computer program Not clear	Not stated	Not stated	Sitting position	Not clear	Program not available	Computer, program
	Naming body parts^Citation48	All children completed the test	Not clear	Computer program Not clear	Not stated	Not stated	Sitting position	Not clear	Program not available	Computer, program

^aoriginal version ‘length of instrument’

^boriginal version ‘completion time’

Abbreviations: TD – typically developing children; y – years; ISNCSCI – International Standard for Neurological Classification of Spinal Cord Injury

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