Health-related quality of life of children treated for non-syndromic craniosynostosis

Figures & data

Table 1. Attrition analysis.

Variables	Non-participating (n = 18)	Participating (n = 73)	P
Sex
Female	6 (33.3%)	24 (32.9%)
Male	12 (66.7%)	49 (67.1%)	1.00
Age at study	11.7 (2.2)	11.0 (2.4)	0.23
	12 (8; 15)	11 (7; 15)
	n = 18	n = 73
Age at surgery (days)	277.1 (338.6)	237.0 (224.7)	0.74
	159.5 (94; 1523)	165 (84; 1484)
	n = 18	n = 73
Craniosynostosis
Sagittal	9 (50.0%)	41 (56.2%)
Metopic	6 (33.3%)	24 (32.9%)
Unicoronal	1 (5.6%)	6 (8.2%)
Bicoronal	1 (5.6%)	1 (1.4%)
Lambdoid	1 (5.6%)	1 (1.4%)	0.64
Surgery method
Pi-plasty	1 (5.6%)	17 (23.3%)
Spring-assisted surgery	8 (44.4%)	23 (31.5%)
Fronto-orbital reshaping with bone graft	6 (33.3%)	14 (19.2%)
Fronto-orbital reshaping with spring	3 (16.7%)	18 (24.7%)
Barrel-stave osteotomy	0 (0.0%)	1 (1.4%)	0.29
Born premature
Yes	3 (17.6%)	5 (6.8%)
No	14 (82.4%)	68 (93.2%)	0.34

For categorical variables, n (%) is presented.

For continuous variables, mean (SD) / median (min; max) / n is presented.

For comparison between groups, Fisher’s Exact test (lowest one-sided.

p-value multiplied by 2) was used for dichotomous variables, a chi-squared test was used for non-ordered categorical variables, and the Mann–Whitney U test was used for continuous variables.

Table 2. Demographics and baseline characteristics.

Variables	Total (n = 73)
Sex
Female	24 (32.9%)
Male	49 (67.1%)
Age at study	11.0 (2.4)
	11 (7; 15)
	n = 73
Craniosynostosis
Sagittal	41 (56.2%)
Metopic	24 (32.9%)
Unicoronal	6 (8.2%)
Bicoronal	1 (1.4%)
Lambdoid	1 (1.4%)
Surgery method
Pi-plasty	17 (23.3%)
Spring-assisted surgery	23 (31.5%)
Fronto-orbital reshaping with bone graft	14 (19.2%)
Fronto-orbital reshaping with spring	18 (24.7%)
Barrel-stave osteotomy	1 (1.4%)
Age at surgery (days)	237.0 (224.7)
	165 (84; 1484)
	n = 73
Involved parent
Mother	36 (50.0%)
Father	12 (16.7%)
Both	24 (33.3%)
Maternal education level
Primary school	2 (3.4%)
High school	21 (35.6%)
College/university	36 (61.0%)
Paternal education level
Primary school	2 (5.7%)
High school	19 (54.3%)
College/university	14 (40.0%)
Born premature
Yes	5 (6.8%)
No	68 (93.2%)
Did the child have other diseases/diagnosis?
Yes	22 (30.6%)
No	50 (69.4%)
What kind of condition?
Heart disease	4 (23.5%)
Neuropsychiatric diagnosis	5 (29.4%)
Asthma/allergy	4 (23.5%)
Migraine	3 (17.6%)
Metabolic disease	1 (5.9%)
Has your child been assessed by a psychologist?
Yes	14 (19.2%)
No	59 (80.8%)
What did the psychological assessment show?
Autism	4 (30.8%)
Problem with executive function	1 (7.7%)
ADHD	2 (15.4%)
Problem with attention	2 (15.4%)
Both autism and ADHD	2 (15.4%)
Development delay	2 (15.4%)
Is your child under medical treatment?
Yes	8 (11.4%)
No	62 (88.6%)
Right- or left-handed?
Right	62 (87.3%)
Left	9 (12.7%)
Wechsler full scale intelligence quotient	98.1 (14.6)
	100 (59; 135)
	n = 72
Wechsler verbal comprehension intelligence quotient	99.5 (14.6)
	100 (57; 130)
	n = 72
Wechsler perceptual reasoning intelligence quotient	104.5 (13.7)
	102 (69; 135)
	n = 72
Wechsler working memory intelligence quotient	94.9 (13.0)
	97 (56; 120)
	n = 71
Wechsler processing speed intelligence quotient	92.1 (14.4)
	91 (53; 126)
	n = 71
Adaptive behaviour skills full scale	94.4 (20.2)
	97.5 (47; 120)
	n = 72
Conceptual composite scale	93.9 (20.1)
	95.5 (40; 119)
	n = 72
Social composite scale	93.4 (19.6)
	97 (42; 118)
	n = 72
Practical composite scale	94.6 (19.8)
	97 (55; 120)
	n = 72

For categorical variables, n (%) is presented.

For continuous variables, mean (SD) / median (min; max) / n is presented.

Table 3. Child-rated PedsQL generic core scales relative to normative sample.

	Craniosynostosis (n = 73)		Sagittal (n = 41)		Metopic (n = 24)
Variables	Mean (SD) Median (Min; Max) n	p (within group)	Mean (SD) Median (Min; Max) n	P (within group)	Mean (SD) Median (Min; Max) n	p (within group)
HRQoL	−0.18 (1.14)	0.20	−0.26 (1.11)	0.14	−0.24 (1.26)	0.45
PedsQL Full Scale	−0.04 (−4.57; 1.24)		−0.09 (−3.49; 1.04)		−0.04 (−4.57; 1.24)
	n = 67		n = 40		n = 21
Physical Function Scale	0.10 (0.91)	0.36	0.09 (0.87)	0.54	−0.02 (1.02)	0.94
	0.14 (−3.80; 1.07)		0.14 (−1.95; 1.07)		0.14 (−3.80; 1.07)
	n = 69		n = 40		n = 23
Emotional Function Scale	−0.06 (1.00)	0.61	−0.10 (0.97)	0.51	−0.09 (1.04)	0.69
	0.09 (−3.00; 1.12)		0.26 (−3.00; 1.12)		0.09 (−3.00; 1.12)
	n = 69		n = 40		n = 23
Social Function Scale	−0.27 (1.31)	0.082	−0.37 (1.35)	0.085	−0.30 (1.36)	0.34
	0.28 (−5.12; 0.70)		0.28 (−5.12; 0.70)		0.28 (−4.29; 0.70)
	n = 69		n = 40		n = 23
School Function Scale	−0.51 (1.32)	0.0014	−0.68 (1.37)	0.0018	−0.43 (1.31)	0.14
	−0.34 (−4.05; 1.14)		−0.34 (−4.05; 1.14)		−0.34 (−4.05; 1.14)
	n = 67		n = 40		n = 23
Psychosocial Function Scale	−0.33 (1.28)	0.035	−0.45 (1.28)	0.031	−0.32 (1.37)	0.32
	0.00 (−4.50; 1.20)		−0.23 (−4.35; 1.20)		−0.15 (−4.50; 1.20)
	n = 67		n = 40		n = 23

For continuous variables, mean (SD) / median (min; max) / n is presented.

For comparison within groups, Fisher’s non-parametric permutation test for matched pairs was used.

For each variable, the z-score was calculated as (PedsQL − normal mean) / normal SD.

Table 4. Difference in the mean of child- and parent-rated PedsQL generic core scales between treated sagittal and metopic CS patients.

Variables	Sagittal (n = 41)	Metopic (n = 24)	p	Adjusted p*	Mean difference with 95% CI^a	Effect size^b
HRQoL	85.4 (9.8)	85.8 (11.2)	0.90		−0.41 (−6.09; 5.05)	0.040
PedsQL Full Scale	86.4 (53.3; 98.4)	87.5 (52.2; 97.8)
	n = 39	n = 21
Physical Function Scale	89.5 (8.0)	87.8 (11.7)	0.53	0.82	1.72 (−3.37; 6.39)	0.181
	90.6 (64.1; 100)	89.1 (39.1; 98.4)
	n = 40	n = 23
Emotional Function Scale	81.7 (11.8)	83.4 (11.8)	0.61		−1.68 (−7.92; 4.42)	0.142
	81.3 (55; 100)	85 (52.5; 97.5)
	n = 40	n = 23
Social Function Scale	89.7 (12.0)	88.4 (14.1)	0.70		1.31 (−5.67; 7.69)	0.102
	92.5 (37.5; 100)	92.5 (42.5; 100)
	n = 39	n = 23
School Function Scale	77.6 (17.7)	83.0 (14.2)	0.24	0.76	−5.41 (−14.55; 3.39)	0.326
	81.3 (35; 100)	87.5 (47.5; 97.5)
	n = 40	n = 21
Psychosocial Function Scale	83.3 (11.9)	84.8 (12.5)	0.66		−1.53 (−8.12; 4.88)	0.127
	85.8 (47.5; 97.5)	86.7 (51.7; 98.3)
	n = 39	n = 21

For continuous variables, mean (SD) / median (min; max) / n is presented.

For comparison between groups, Fisher’s non-parametric permutation.

test was used for continuous variables.

^aData represent the mean (95% CI). The CI for the difference between groups is based on Fisher’s non-parametric.

permutation test.

^bData represent the difference in the mean / pooled SD.

* Adjustment of physical function for age at study and school function for WMIQ was performed by ANCOVA.

CI: confidence interval.

Table 5. Difference in the mean of child- and parent-rated PedsQL generic core scales between pi-plasty and spring-assisted surgery used to treat patients with sagittal CS.

Variables	Pi-plasty (n = 17)	Spring-assisted surgery (n = 23)	p	Difference between groups^a	Effect size^b
HRQoL	84.9 (11.7)	85.6 (8.6)	0.83	−0.74 (−7.41; 5.98)	0.074
PedsQL Full Scale	88.3 (53.3; 97.8)	86.4 (67.4; 98.4)
	n = 16	n = 22
Physical Function Scale	89.2 (10.0)	90.1 (6.4)	0.75	−0.90 (−6.25; 4.49)	0.111
	89.1 (64.1; 100)	91.4 (76.6; 100)
	n = 17	n = 22
Emotional Function Scale	82.1 (13.6)	81.4 (10.8)	0.89	0.70 (−7.19; 8.75)	0.057
	82.5 (55; 100)	80 (57.5; 95)
	n = 17	n = 22
Social Function Scale	87.2 (15.0)	91.1 (9.4)	0.38	−3.95 (−11.79; 4.06)	0.328
	92.5 (37.5; 100)	92.5 (65; 100)
	n = 16	n = 22
School Function Scale	77.4 (18.0)	77.3 (18.2)	1.00	0.08 (−11.67; 12.19)	0.004
	80 (42.5; 95)	81.3 (35; 100)
	n = 17	n = 22
Psychosocial Function Scale	82.9 (13.2)	83.3 (11.4)	0.94	−0.34 (−8.42; 7.81)	0.028
	86.7 (47.5; 96.7)	84.2 (54.2; 97.5)
	n = 16	n = 22

For continuous variables, mean (SD) / median (min; max) / n is presented.

For comparison between groups, Fisher’s non-parametric permutation.

test was used for continuous variables. .

^aData represent the mean (95% CI). The CI for the difference between groups is based on Fisher’s non-parametric.

permutation test.

^bData represent the difference in the mean / pooled SD.

CI: confidence interval.

Table 6. Correlation analysis of the mean of child- and parent-rated PedsQL generic core scales with ABAS and IQ.

Variables	HRQoL PedsQL Full scale	Physical Function Scale	Emotional Function Scale	Social Function Scale	School Function Scale	Psychosocial Function Scale
Adaptive behaviour skills full scale	0.57	0.32	0.50	0.40	0.55	0.57
	<0.0001	0.0084	<0.0001	0.0008	<0.0001	<0.0001
	65	68	68	67	66	65
Conceptual composite scale	0.57	0.33	0.46	0.38	0.57	0.57
	<0.0001	0.0053	<0.0001	0.0013	<0.0001	<0.0001
	65	68	68	67	66	65
Social composite scale	0.52	0.38	0.52	0.35	0.44	0.52
	<0.0001	0.0015	<0.0001	0.0038	0.0002	<0.0001
	65	68	68	67	66	65
Practical composite scale	0.53	0.28	0.46	0.37	0.52	0.53
	<0.0001	0.022	<0.0001	0.0020	<0.0001	<0.0001
	65	68	68	67	66	65
Wechsler full scale intelligence quotient	0.42	0.16	0.28	0.34	0.47	0.42
	0.0004	0.19	0.022	0.0046	<0.0001	0.0004
	65	68	68	67	66	65
Wechsler verbal comprehension intelligence quotient	0.39	0.26	0.26	0.30	0.40	0.39
	0.0014	0.030	0.036	0.013	0.0009	0.0014
	65	68	68	67	66	65
Wechsler perceptual reasoning intelligence quotient	0.31	0.05	0.18	0.22	0.34	0.31
	0.013	0.66	0.13	0.069	0.0047	0.013
	65	68	68	67	66	65
Wechsler working memory intelligence quotient	0.20	0.06	0.14	0.16	0.29	0.20
	0.12	0.60	0.27	0.20	0.019	0.12
	65	68	68	67	66	65
Wechsler processing speed intelligence quotient	0.36	−0.01	0.19	0.33	0.43	0.36
	0.0030	0.93	0.12	0.0065	0.0003	0.0030
	65	68	68	67	66	65

For each variable, Spearman’s correlation is presented with the corresponding p-value and the number of observations used.

Table 7. Change in PedsQL generic core scales from child rater to parent rater.

	Child (n = 73)	Parent (n = 73)	Change from child to parent (n = 73)
Variable	Mean (SD) Median (Min; Max) (95% CI for the mean), n	Mean (SD) Median (Min; Max) (95% CI for the mean), n	Mean (SD) Median (Min; Max) (95% CI for the mean), n SRM ES	p (within group)
HRQoL	84.3 (12.5)	88.0 (11.4)	3.61 (13.15)	0.031
PedsQL full scale	85.9 (35.9; 100)	90.8 (58.7; 100)	3.8 (−19.57; 33.7)
	(81.3; 87.3)	(85.2; 90.7)	(0.40; 6.84)
	n = 67	n = 68	n = 66
			0.27
			0.29
Physical function scale	87.0 (12.2)	92.2 (10.9)	5.25 (13.53)	0.0022
	87.5 (34.4; 100)	96.9 (43.8; 100)	3.13 (−21.88; 37.5)
	(84.0; 89.8)	(89.6; 94.8)	(2.00; 8.52)
	n = 69	n = 69	n = 69
			0.39
			0.43
Emotional function scale	82.8 (14.5)	82.5 (15.4)	−0.290 (18.921)	0.94
	85 (40; 100)	85 (40; 100)	0 (−40; 60)
	(79.3; 86.2)	(78.9; 86.2)	(−4.833; 4.242)
	n = 69	n = 69	n = 69
			−0.02
			−0.02
Social function scale	88.3 (15.8)	91.4 (13.2)	2.87 (15.34)	0.14
	95 (30; 100)	97.5 (35; 100)	0 (−30; 45)
	(84.6; 92.1)	(88.2; 94.6)	(−0.78; 6.56)
	n = 69	n = 68	n = 68
			0.19
			0.18
School function scale	77.7 (17.8)	83.0 (19.6)	5.82 (18.14)	0.012
	80 (30; 100)	90 (30; 100)	10 (−50; 60)
	(73.4; 82.0)	(78.4; 87.7)	(1.41; 10.30)
	n = 67	n = 69	n = 67
			0.32
			0.33
Psychosocial function scale	83.0 (14.2)	85.8 (13.7)	2.60 (14.98)	0.17
	86.7 (36.7; 100)	90 (46.7; 100)	3.33 (−33.33; 45)
	(79.5; 86.4)	(82.5; 89.1)	(−1.03; 6.26)
	n = 67	n = 67	n = 66
			0.17
			0.18

For continuous variables, mean (SD) / median (min; max) / (95% CI using the inversion of Fisher’s non-parametric permutation test) / n is presented.

For comparison within groups, Fisher’s non-parametric permutation test for matched pairs was used.

Change was calculated, as follows: difference = parent rate − child rate.

SRM, standardized response mean = mean difference / SD of the difference.

ES, effect size = mean difference / SD for child.

CI: confidence interval.