The Type, Severity, and Impact of Sleep Problems in Children With Angelman Syndrome and Parental Help-seeking Patterns

Figures & data

Table 1. Demographic characteristics of the sample.

Demographic data	N, Percentage of total sample
Parent age
18–24	2, 1.5%
25–34	32, 23.0%
35–44	69, 49.6%
45+	32, 23.2%
Did not report	4, 2.90%
Age of child
0–4	51, 40.0%
5–10	43, 31.2%
11–14	20, 14.5%
15–18	14, 10.1%
18+	8, 5.8%
Relationship to child
Mother	112, 81.2%
Father	25, 18.1%
Other	2, 1.5%
Ethnicity
European/Caucasian/Pakeha/White	98, 71.0%
Asian	7, 5.1%
Latino/Hispanic	7, 5.1%
Other	22, 15.9%
Did not report	5, 3.6%
Marital status
Married	122, 88.4%
Single, never married	4, 2.9%
Divorced or separated	12, 8.7%
Widowed	1. 0.7%
Household size
2–3	23, 16.5%
4–5	108, 77.7%
6+	7, 5.0%
Employment
Home maker	22, 15.8%
Employed for wages	87, 62.6%
Self-employed	10, 7.2%
Unemployed	16, 11.5%
Student	2, 1.5%
Did not report	2, 1.5%
Highest degree completed
Postgraduate	60, 43.1%
Undergraduate	37, 26.6%
Finished high school	39, 28.1%
Did not finish high school	3, 2.2%

Table 2. Practices used by parents, ratings of effectiveness, and *level of empirical support in children with Angelman syndrome or other developmental disabilities and sleep disturbance.

	Used		Rated as effective		Resolved sleep problem
	n	% of total P	n	% of P used	N	% of P used
Supported
**Prescription medication	118	85	81	69	3	3
Systematic ignoring	100	72	19	19	1	1
Changing bedroom environment (e.g., removing toys, reducing noise)	90	65	36	40	1	1
Bedtime fading	82	59	33	40	2	2
Modified systematic ignoring	78	56	18	23	2	3
Scheduled awakening	38	27	10	26	1	3
CPAP	12	9	4	33	0	0
Some support
White noise	91	65	44	48	2	2
Social stories	65	47	12	18	1	2
Video modeling	38	27	5	13	1	3
Unsupported
Co-sleeping	104	75	70	67	1	1
Weighted blanket	71	51	27	38	0	0
Exercise	89	64	55	62	1	1
Homeopathic	67	48	21	31	0	0
Massage	65	47	25	37	0	0
Other
Surgery	17	12	5	29	1	6
Other – unspecified	11	8	5	45	1	9

CPAP – continuous positive airway pressure.

*Classifications of empirical support are based on evidence from systematic reviews and meta-analyses (e.g., Carnett et al., 2019; France et al., 2018; Hollway & Aman, 2011; McLay & France, 2016; Turner & Johnson, 2013). Supported techniques had sound empirical support; those with some support had a sound theoretical basis and extant studies showing limited evidence of effects; unsupported techniques lacked empirical evidence of effectiveness with children with developmental delays.

**Almost half were taking melatonin while a small number were taking CBD oil. Other medications reported for sleep treatment were clonidine, anti-convulsant medications, anti-depressants such as trazadone, and small numbers reported using antihistamines, and antipsychotic medications (e.g., risperidone).

P = participant/s.

Table 3. Parent-reported sleep problems and sleep-related medical problems.

Sleep problem	N	Percentage total sample
Insomnia	139	100
Frequent night wakings	98	71
Early morning wakings	87	63
Sleep onset delay	85	61
Prolonged night wakings	85	61
Co-sleeping – child’s bed	37	27
Co-sleeping – parent’s bed	32	23
Daytime sleepiness	11	8
Circadian rhythm disturbances	91	66
Fragmented sleep	47	34
Sleep phase disturbance	45	32
Biphasic sleep pattern	33	24
Delayed sleep phase	24	17
Advanced sleep phase	21	15
Polyphasic sleep pattern	14	10
Sleep-interfering behavior	54	39
Frequent bids for parental attention	37	27
Leaving bed	26	19
Playing with toys	26	19
Bedtime resistance	24	17
Repetitive motor movements	23	17
Crying or upset	22	16
Vocalizing to self	16	12
Non-compliance with the bedtime routine	16	12
Nighttime laughing	11	8
Other	19	14
Parasomnias	26	19
Teeth grinding	17	12
Night terrors/nightmares	7	5
Sleep talking	3	2
Sleep walking	2	1
Other	6	4
Other
Unexplained crying	24	17
Medical problems	97	70
Gastrointestinal problems	52	37
Seizure disorder	45	32
Unusual nighttime movements (unexplained, while asleep)	37	27
Bedwetting	37	27
Identified problems with endogenous melatonin	35	25
Sleep-related breathing problems (e.g., OSA)	13	9
Eczema	13	9
Asthma	7	5
Other (unspecified)	13	9

OSA – Obstructive sleep apnea.

Table 4. Parental concern, importance to treat, and child and family impacts of sleep problems.

	N	Percentage of sample
Parental levels of concern
Most significant	25	19
Very concerned	51	38
Somewhat Concerned	44	33
Mildly concerned	13	10
Not concerned	1	75
Important to treat
My primary focus	42	30
Very Important	59	42
Somewhat important	23	17
Mildly important	7	5
Impact on child
Daytime behavior-home	75	51
Daytime behavior school	67	48
Learning	58	42
Emotional regulation	57	41
Communication	38	27
Social interaction	36	26
Relationships	13	9
Impact on family
Parent sleep	111	80
Spouse sleep	75	54
Parental daytime functioning	93	67
Parental mental health	89	64
Parental employment	69	50
Spousal relationship	70	50
Physical wellbeing	66	47
Sibling sleep	44	32
Sibling relationship	37	27
Financial security	12	9

Table 5. Sources of information and support about child’s sleep problems, advice received, and quality of support.

	N	Percentage of sample
Professional support	88	63
Medical professional	77	55
Psychologist	11	8
Occupational therapist	10	8
Board certified behavior analyst	11	8
Speech language pathologist	2	1
Researcher	4	3
Physical therapist	2	1
Quality of support
Suitable or extensive/regular support	28	20
Little or occasional support	82	59
No support	22	14
Professional advice reported
Feature of AS	61	44
Use medication	27	19
Treatment required	7	5
Children can learn to improve sleep	7	5
Sleep problem will respond to environmental modifications	3	2
Sleep problem likely to resolve without treatment	3	2
Other parent-initiated resources	68	49
Web-based supports	35	25
Other parents	30	22
Books	17	5
Other	18	13

Carnett, A., Hansen, S., McLay, L. K., Neely, L., & Lang, R. (2019). Quantitative-analysis of behavioral interventions to treat sleep problems in children with autism. Developmental Neurorehabilitation, 23(5), 271–284. https://doi.org/10.1080/17518423.2019.1646340

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