Sleep and Its Disturbance in Parents of Children and Adolescents with Epilepsy: A Systematic Review and Meta-Analysis

Figures & data

Figure 1 Preferred reporting items for systematic reviews and meta-analyses (PRISMA) flow diagram for study selection and exclusion.

Notes: PRISMA figure adapted from Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71. Creative Commons.²⁴

Table 1 Study Participant Characteristics of the Included Studies

Source, Year, Country	Sample Size (% of Mother)	Parental Age (Yr) Mean (SD)	Children’s Age (Yr)	Sleep Measure	Sleep Variable and Result	Quality^a
Cross-sectional study
Cottrell et al, 2005, USA^Citation16	E: 50 (100) C: NA	E: 32.3; range 21–39 C: NA	E: < 5 C: NA	PSQI	Nocturnal sleep duration: 4.5 hours Nighttime waking: 3 times/night Sleep problem: 59.9%	3/8
Liu et al, 2021, China^Citation6	E: 157 (57.3) C: NA	E: 37.14 (5.57); range 25–54 C: NA	E: < 18 C: NA	PSQI	PSQI total score: 6.41 (SD=3.79) PSQI total score ≥ 7: 37.6%	8/8
Tsai et al, 2020, Taiwan^Citation18	E: 133 (100) C: NA	E: 35.82 (4.32); range 23–46 C: NA	E: 1.5–6 C: NA	PSQI	PSQI total score: 6.21 (SD=3.17) PSQI total score > 5: 57.1% Sleep latency ^b: 25.0 (10.0, 30.0) minutes Nocturnal sleep duration: 6.57 (SD=1.45) hours Nocturnal sleep duration < 6 hours: 25.6% Sleep efficiency: 86.86 (SD=13.89) % Sleep efficiency < 85%: 33.8%	8/8
Wood et al, 2008, Canada^Citation15	E: 52 (100) C: NA	E: NR C: NA	E: 2–18 C: NA	PSQI	PSQI total score: 7.7 PSQI total score > 5: 67%	7/8
Case-control study
Larson et al, 2012, USA^Citation23	E: 105 (94.3) C: 79 (88.6)	E: NR C: NR	E: 2–10 C: 2–10	PSQI	Epilepsy vs Control PSQI total score*: 6.37 (SD=3.49) vs 4.67 (SD=3.21) PSQI daytime dysfunction subscore*: 1.07 (SD=0.78) vs 0.67 (SD=0.76) PSQI habitual sleep efficiency subscore*: 0.67 (SD=0.98) vs 0.37 (SD=0.80) PSQI sleep latency subscore: 0.93 (SD=0.97) vs 0.63 (SD=0.93) PSQI subjective sleep quality subscore: 1.34 (SD=0.83) vs 1.09 (SD=0.08) PSQI use of sleep medication: 0.33 (SD=0.79) vs 0.11 (SD=0.42) PSQI Sleep disturbances subscore*: 1.24 (SD=0.52) vs 1.10 (SD=0.49) PSQI Sleep duration subscore: 0.81 (SD=0.86) vs 0.70 (SD=0.95)	9/10
Painter et al, 2014, USA^Citation29	E: 30 (86.6) C: 29 (100)	E: NR C: NR	E: 2–12 C: 2–12	Daily phone diary	Time spent in sleep: longer on the weekends versus the weekdays (41.66 minutes, p < 0.01).	10/10
Reilly et al, 2018, UK^Citation7c	E: Mother 47; Father 39 C: Mother 48; Father 42	E: Mother 35.29; Father 37.95 C: Mother 33.71; Father 37.61	E: 1–7 C: 1–7	PSQI	Epilepsy PSQI total score (mother): 7.91 (SD=4.28) PSQI total score (father): 6.36 (SD=3.9) PSQI total score > 5 (mother): 62% PSQI total score > 5 (father): 44% Nonepilepsy-related neurodisability PSQI total score (mother): 7.35 (SD=4.31) PSQI total score (father): 5.71 (SD=3.05) PSQI total score > 5 (mother): 60% PSQI total score > 5 (father): 55%	9/10
Shaki et al, 2011, Israel^Citation17	E: 39 (66.6) C: 42 (57.1)	E: 39.92 (7.29) C: 36.43 (7.29)	E: < 18 C: < 18	PSQI	Epilepsy vs Control Nocturnal sleep duration: 5.26 (SD=1.55) vs 6.04 (SD=1.27) hours PSQI total score > 5: OR = 6.66, CI 95% (1.10–70.08)	9/10
Yang et al, 2020, China^Citation5	E: 234 (67.5) C: 230 (66.9)	E: 37.56 (6.94) C: 39.02 (6.33)	E: < 18 C: < 18	PSQI	Epilepsy vs Control PSQI total score (all)*: 6.94 (SD=3.81) vs 5.03 (SD=3.39) PSQI total score (infant)*: 9.02 (SD=4.34) vs 4.93 (SD=3.74) PSQI total score (preschooler): 6.57 (SD=3.42) vs 5.98 (SD=4.35) PSQI total score (schooler)*: 6.14 (SD=3.46) vs 4.18 (SD=2.06) PSQI total score (adolescent)*: 5.84 (SD=2.96) vs 4.18 (SD=2.06)	9/10
Cohort study
Borusiak et al, 2016, Germany^Citation30	E: 36 (NR) C: NA	E: NR C: NA	E: 1–16 C: NA	Single item from PSQI	Sleep quality: improved from diagnosis to 5–7 months of follow-up (p = 0.03).	8/11
Quasi-experimental study
Ledet et al, 2016, USA^Citation31	E: 12 (91.7) C: NA	E: 35.75 (6.62); range 25–49 C: NA	E: 2–17 C: NA	PSQI ESS	PSQI total score (pretest): 10.55 (SD=3.75) PSQI total score (posttest): 10.09 (SD=3.61) PSQI total score (pretest) > 5: 100% PSQI total score (posttest) > 5: 83% ESS total score (pretest): 10.58 (SD=3.82) ESS total score (posttest): 10.58 (SD=4.73) ESS total score (pretest) > 10: 58% ESS total score (posttest) > 10: 58%	7/9

Notes: ^aQuality = Joanna Briggs Institute quality appraisal number of items with yes/total number of items; ^bMedian and interquartile range were reported; ^cChildren with nonepilepsy-related neurodisability formed the control group; *Significant differences between the epilepsy and control groups (p < 0.05).

Abbreviations: CI, confidence interval; C, control; controls were formed by healthy children, unless otherwise indicated; E, epilepsy; ESS, Epworth Sleepiness Scale; NA, not applicable; NR, not reported; OR, odds ratio; PSQI, Pittsburgh Sleep Quality Index; SD, standard deviation.

Figure 2 (A) Forest plot of the mean for Pittsburgh Sleep Quality Index (PSQI) total score in parents of children and adolescents with epilepsy; (B) funnel plot of studies estimating PSQI total scores in parents of children and adolescents with epilepsy, adjusted for unpublished reports (black dots).

Table 2 Subgroup Analysis and Meta-Regression of Sleep Quality and Prevalence of Parental Sleep Disturbance

Variables	Study No.	Pooled Estimate	95% CI	p
Sleep quality (n=7)
Country				0.070
Western^a	4	7.87	6.49 to 9.25
Eastern^b	3	6.53	6.08 to 6.98
Sample size				0.063
≤ 50	2	9.08	6.51 to 11.65
> 50	5	6.63	6.21 to 7.05
Sample size	7	β = −0.010	−0.021 to −0.001	0.452
Child age	4	β = 0.37	−0.61 to 1.37	0.456
Study quality score	7	β = −0.09	−0.14 to −0.03	< 0.001
Prevalence of sleep disturbances (n=6)
Country				0.115
Western^a	4	0.64	0.54 to 0.72
Eastern^b	2	0.47	0.29 to 0.65
Sample size				0.351
≤ 50	3	0.64	0.48 to 0.77
> 50	3	0.53	0.36 to 0.69
Sample size	6	β = −0.008	−0.014 to −0.002	0.007
Child age	6	β = 0.01	−0.20 to 0.22	0.924
Study quality score	6	β = −0.01	−0.02 to 0.01	0.453

Note: ^aIndicates USA, UK, and Canada; ^bIndicates China and Taiwan.

Figure 3 (A) Forest plot of the overall prevalence of sleep disturbances in parents of children and adolescents with epilepsy; (B) funnel plot of studies estimating the prevalence of sleep disturbances in parents of children and adolescents with epilepsy.

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