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Comprehensive Summaries of Uppsala Dissertations from the Faculty of Medicine 1108 _____________________________ _____________________________ Severe Sleep Problems among Infants A Five-Year Prospective Study BY MALENA THUNSTRÖM ACTA UNIVERSITATIS UPSALIENSIS UPPSALA 2002

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Comprehensive Summaries of Uppsala Dissertationsfrom the Faculty of Medicine 1108

_____________________________ _____________________________

Severe Sleep Problemsamong Infants

A Five-Year Prospective Study

BY

MALENA THUNSTRÖM

ACTA UNIVERSITATIS UPSALIENSISUPPSALA 2002

Dissertation for the Degree of Doctor of Philosophy (Faculty of Medicine) in Paediatricspresented at Uppsala University in 2002

ABSTRACT

Thunström, M. 2002. Severe Sleep Problems among Infants. A Five-Year ProspectiveStudy. Acta Universitatis Upsaliensis. Comprehensive Summaries of Uppsala Dissertationsfrom the Faculty of Medicine 1108. 77 pp. Uppsala. ISBN 91-554-5201-9.

The aim of this thesis was to explore the prevalence of parentally experienced infant sleepproblems, with special interest in severe problems, in a total community sample of 2 518infants aged between 6 and 18 months. Factors associated with severe sleep problems weresought. Parents reported 16 % of the infants to have difficulties in falling asleep at night,and 30 % to have frequent night waking. Severe sleep problems were associated withfrequent night meals, psychosocial problems in the family, exhaustion and depression inthe mother, and parental stress. An association with infant difficultness, high activity andproblematic behaviour was also found.

In a five-year prospective study a group of children fulfilling specific criteria for severesleep problems in infancy (N=27) was followed after an interventional sleep programmeand compared with a control group regarding sleep characteristics, behaviour anddevelopment. One month after an interdisciplinary treatment programme, combiningbehavioural technique with family work, the average number of times the case babies wokeup had diminished from 6.0 to 1.8 times per night. A 92 % rate of improvement wasreported.

The changes were stable over time. Comparisons with the controls during five yearsrevealed no significant group difference in sleep characteristics. Concerning behaviour anddevelopment, however, there were significant differences. At the age of 5.5 years, seven ofthe children in the former sleep problem group met the criteria for the diagnosis ofattention-deficit/hyperactivity disorder. No control child qualified for the diagnosis.

Close follow-ups of infants with combined severe sleep and behavioural problems are recommended.

Key words: Sleep disorders, infant, behavioural therapy, psychosocial problems, attention-deficit/hyperactivity disorder.

Malena Thunström, Department of Women´s and Children´s Health, Uppsala UniversityChildren´s Hospital, SE-751 85 Uppsala, Sweden

© Malena Thunström 2002

ISSN 0282-7476ISBN 91-554-5201-9

Printed in Sweden by Uppsala University, Tryck & Medier, Uppsala 2002

To all sleepy children

LIST OF PAPERSThis thesis is based on the following papers, which will be referred to in thetext by their Roman numerals:

I Thunström M.Severe sleep problems among infants in a normal population in Sweden:prevalence, severity and correlates. Acta Paediatrica 1999; 88: 1356-1363.

II Thunström M.Severe sleep problems among infants: family and infant characteristics.Ambulatory Child Health 1999; 5: 27-41.

III Thunström M.A 2.5-year follow-up of infants treated for severe sleep problems.Ambulatory Child Health 2000; 6: 225-235.

IV Thunström M.Severe sleep problems in infancy associated with subsequentdevelopment of attention–deficit/hyperactivity disorder at 5.5 years ofage. Acta Paediatrica. Accepted for publication.

V Thunström M.A 5-year follow-up of infants with severe sleep problems. Manuscript.

Reprints were made with the permission of the publishers.

CONTENTS

INTRODUCTIONGeneral background 7Maturation of sleep pattern in childhood 7Definition and classification of sleep disorders 9Assessment of sleep 11Epidemiology of sleep problems:prevalence, severity and persistence 12Correlates to disturbed sleep 14Treatment of sleep problems in childhood 15Attention-deficit/hyperactivity disorder (ADHD) and sleep 16Aims of the studies 21

METHODSDesign and subject selection 22 Study I 22 Study II-IV 22 Study V 24Procedures and measures 24

Study I 24Questionnaire on sleep and background factors 24Study II, III, V 25Questionnaires on sleep, family and infant characteristics and sleep diaries. 26Treatment programme 28Study IV 30Screening instrument for ADHD 30Assessment methods 31

Statistics 33

RESULTSPrevalence and classification of sleep problems 33Family and infant characteristics associated with severe infant sleep problems 36Short-term and long-term outcome of an interdisciplinary sleep treatment programme 44Factors associated with subsequent development of attention-deficit/hyperactivity disorder 48

DISCUSSIONGeneral discussion 52Validity of the results 58Conclusions and clinical implications 61ACKNOWLEDGEMENTS 63REFERENCES 65

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INTRODUCTION

General background

Sleep plays a major role in children´s well-being and is strongly influenced by

the child´s health status, psychological stress and family issues as well as by

multiple aspects of his or her culture and environment. Also, children´s sleep

patterns affect their well-being within the same wide range of health and

psychosocial phenomena.

Research has shown that sleep problems in infants and young children are

prevalent. Cross-sectional studies show that between 15 % and 35 % of all

children aged between 6 months and 5 years are reported by their parents to

show some type of sleep disturbance (22,69,76,98,100,102).

Before six months of age it is doubtful whether the label "sleep disturbance"

should be used, since during this period night waking is necessary in order to

fulfil the needs of frequent nutrition of the child. This is also the period

needed to reorganize the sleep pattern of the newborn to a more mature

pattern (85).

Maturation of normal sleep pattern in childhood

Sleep and sleep stages are defined by specific patterns of electro-

physiologically recorded brain activity, muscle tone and eye movements.

These polysomnographic measures divide sleep into two principal states:

sleep associated with rapid eye movements, "REM sleep", and sleep without

rapid eye movements, "non–REM sleep" (28). The REM component of sleep,

which dominates during the foetal period and the early months of life (50 %

of the sleep time), is thought to play a crucial role with regard to brain

8

development (105). The amount of REM sleep decreases to 30 % during the

first year of life and the REM–non REM state cycle also undergoes

maturational changes. In newborns and very young infants the cycle lasts

approximately 40 to 60 minutes. At two years of age the cycle has increased

to 75 minutes and continues to increase to an average of 84 minutes in 5-year-

olds (65).

A newborn baby does not distinguish day from night. Both sleeping and

eating take place around the clock, equally distributed over a 24-hour-period,

and the two processes are linked in time to each other. The baby wakes up to

eat and most often goes back to sleep while suckling and being fed at the

breast.

Gradually, in the course of the baby´s first year, a transition to a more regular

pattern of eating and sleeping takes place (10). The child acquires a pattern of

regular meals during daytime, a period of prolonged sleep during the night

and one, or most often two, short naps during daytime. Eating and sleeping

are increasingly separated, and, normally, the one-year-old child has grown

out of the habit of going to sleep while eating.

Temporary nightwaking in occasional periods of short duration is very

common among infants (42), but in the absence of any current physical

disease, a normal development and maturing of the central nervous system

(10) and normal parental handling (10,125), 90 % of one-year-olds sleep all

night (85). Prolonged and complicated sleep problems, however, have a

tendency to remain, especially from the first year of life to later childhood

(1,21,67,92). Moreover longitudinal studies (20,41,60,115) have indicated

that the attitudes and (over-) responsiveness of the parents to the signals of the

child in the parent–infant interaction could play a role in the maintenance of

the sleep problems of the child.

9

Other possible causative and maintaining mechanisms that have been studied

include factors in the genetic, somatic, psychological and psychosocial areas

(19-20,32,61,81,89). Prolonged, complicated sleep problems show a picture

of multiproblem, where the separate factors seem to amplify each other in

vicious circles. Factors affecting the child, the stressed and exhausted parents

and the social situation probably influence each other reciprocally and

interfere with the normal maturation of the sleep pattern.

Definition and classification of sleep disorders

Sleep problems in childhood are classified according to the International

Classification of Sleep Disorders: Diagnostic and Coding Manual (ICSD,

1990, American Sleep Disorder Association) into the following categories:

1. Dyssomnia

Dyssomnia comprises disorders that cause either insomnia (most common) or

excessive sleepiness (rare). Childhood insomnia is most often seen in limit-

setting sleep disorder (in which inadequate enforcement of bedtimes by the

parent results in the child stalling or refusing to go to bed at an appropriate

time) and in sleep-onset association disorder (in which the onset of sleep in

the child is impaired by the absence of a certain object or set of

circumstances). The first conditions is often called "sleep refusal" or "bedtime

struggles" ( a vivid expression) and has been reported in 5-10 % of a normal

population of preschool children (44). The second condition is often called

"frequent night-waking", when the child needs parental intervention of some

form several times every night to go back to sleep, and is seen in 15-20 % of

all children between 6 months and 3 years of age (44,98). Severity is

systematically classified along an axis in the ICSD, 1990, but this

classification and coding system has not been widely used in studies.

10

The ICSD criteria of severe sleep refusal (limit-setting sleep disorder) require

five or more episodes per night of stalling, calling out or leaving the bedroom.

The ICSD criteria of severe night waking problems (sleep-onset association

disorder) require a prolonged sleep latency and over three nightly wakings, or

two or three wakings, each lasting over 10 minutes or one lasting over 15

minutes. The awakenings occur at least five nights per week. Corresponding

detailed definitions for mild and moderate forms of the different sleep

disturbances are also found in the ICSD.

Nocturnal eating syndrome and schedule disorders are other types of

insomnia. Nocturnal eating (drinking) syndrome is characterized by recurrent

awakenings, with the inability to return to sleep without eating or drinking,

preferably breastmilk or formula. In schedule disorder the distribution of

sleep in terms of nap frequency and length across a 24-hour period, the sleep-

wake cycle, is altered in a way that comes into conflict with social

expectations and habits. The child´s sleep occurs at an inappropriate time.

Of course these definitions are cultural, as are the expectations of what is

"normal" infant sleep. Breast-fed babies throughout the world wake at night

and fall asleep at the breast, and in industrialized societies, in which the ICSD

1990 was written, we may have established inappropriate expectations from

experience with bottle-fed infants.

Nevertheless, parents of children with the above-mentioned sleep patterns

report themselves to be exhausted and in need of paediatric help.

Paediatricians, therefore, must have a good knowledge of infant sleep and

factors related to severe problems.

Excessive sleepiness is seen in the rare disorder, narcolepsy, and more often

in the obstructive sleep apnea syndrome.

11

2. Parasomnia

Parasomnia comprises disorders that intrude into or occur during sleep that

are not, primarily disorders of the states of sleep and wakefulness per se.

Included here are confusional arousals, sleepwalking, sleep terrors, sleep

talking and sleep bruxism. Parasomnia is seen predominantly in children aged

3-12 years, where it encompasses 1-6 % of the population (7).

3. Medical/Psychiatric Sleep Disorders

Medical/Psychiatric Sleep Disorders comprise the medical and psychiatric

disorders commonly associated with sleep disturbance. In acute and chronic

illness (123), above all in diseases with pains or itching sensations, (38) sleep

problems are common, but the most difficult and prolonged sleep problems

are seen in mentally retarded children and children with autism, indicating a

common factor of dysfunction of the central nervous system (16).

Assessment of sleep

Clinical assessment of sleep disorders in children includes developmentally

appropriate history-taking of sleep-wake behaviour, including medical,

psychiatric as well as family and psychosocial issues (8,42,84). Physical

examination should be performed, with special attentiveness to signs of

diseases accompanied by pains, breathing difficulties or itching sensations.

Questionnaires and sleep logs or sleep diaries are valuable tools in the process

of detailed history-taking concerning sleep-wake complaints (8,121).

Actigraphy was developed as a practical measure for long-term quantification

of sleep/wake periodicity of movement, since both infants and children have a

lower activity level in sleep than in wakefulness. A small motion sensor and

12

digital recorder is worn on a wrist and later downloaded into a computerized

analysis system. Advantages include noninvasiveness, ease of use and

automated scoring. Accuracy for sleep-wake discrimination has been reported

to be 85-93 % (111-112).

Audio and video recordings as well as polysomnography or other specialized

laboratory test are seldom used in infant studies. In clinical practice, selected

complicated cases are referred to sleep clinics with extended laboratory

devices (multichannel recordings, cardiorespiratory video systems, pulse

oximetry) (24,58).

Results from studies comparing parental reports with objective sleep

measures (109) show that parents are accurate reporters of sleep schedule

measures such as sleep onset and sleep duration. They overestimate, however,

the time that their infants spend in actual sleep and underestimate the number

of their night wakings. According to that result, short sleep duration and many

night wakings reported by parents of small infants should be regarded as a

minimum rather than be suspected of being an exaggeration by exhausted

care-takers.

Epidemiology of sleep problems: prevalence, severity

and persistence

Sleep disturbances in infants and young children, such as night waking and

bedtime struggles, are among the most common behaviour problems

encountered in paediatric practice. Night waking occurs in 15 % to 35 % of

children aged between 6 months and 5 years (17-18,21,59,69,76,94,98,102)

and the prevalence of bedtime struggles varies from 6 % to 16 % in the first 2

years (18,76,94) according to previous studies.

13

These earlier studies show that approximately one preschool child out of four

shows some kind of sleep problem, most often of short duration. A small

group of children, however, approximately 5 %, have more longlasting and

complicated sleep disturbances that can have an influence both on the health

of the child and on the function of the whole family.

Information on the long-term course of sleep problems in children (33,70-71),

and particularly in preschool children, is sparse. Richman, in her survey of

cross-sectional and longitudinal studies of disorders of initiating and

maintaining sleep (100), concludes that although correlations for waking

problems at different ages are reported low in schoolchildren (70), a

considerable number of preschool children have persistent sleep problems.

Especially during the first 2 to 3 years, the probability of continued waking is

high.

Infants who are irritable, sleep little and cry frequently during the early weeks

of life are more likely to be poor sleepers during the second year of life

(20,23). One survey (60) found that just under half (44 %) of those children

waking regularly at 6 months were still waking at 1 year. Furthermore almost

the same proportion (41 %) of the night wakers at 1 year were still waking at

18 months, and from 18 months to 2 years just over half of the children (54

%) continued waking. Conversely, 80 % to 90 % of nonwakers at these ages

continued to sleep well. Continuity after 2 years was less marked: 71 % of

night wakers at 2 years of age slept well at 3 years of age, and 86 % of wakers

at 3 slept well at 4.5 years of age.

Another community survey (103) found more problems. Of those with

bedtime problems at 3 years of age, 25 % still had problems at 4 years of age.

For night waking problems the figure was 36 %. Persistence of the same

problems in children 4 to 8 years of age was 40 % and 17 %, respectively.

14

There was a small core of persistent wakers, who Richman (100) speculates

could be particularly susceptible to irregular sleep patterns because of

neurophysiological or temperamental factors.

In another study (129) children with persistent sleep problems from 8 months

to 3 years of age (41 %), were found to be more likely to have behaviour

problems, especially temper tantrums and behaviour management problems,

than were children without persistent sleep problems. Finally, 84 % of sleep-

disturbed two-year-olds still had persistent sleep disturbances 3 years later in

a study (67) that found that persistent sleep disturbances had a significant

relationship with increased frequency of stress factors in the environment.

Different definitions of night waking and bedtime problems in different

studies contribute to generating different prevalence, severity and persistency

figures, but in summary, longlasting and complicated sleep problems seem to

have a tendency to remain, especially from the first year of life to later

childhood (1,21,67,92) and to be associated with behavioural problems

(119,129) as well as extreme tiredness and stress in the family (1,13,51,53).

Correlates to disturbed sleep

Factors in the genetic, somatic, psychological and psychosocial areas

correlate to disturbed sleep, as possible causative or maintaining mechanisms.

Genetic vulnerability, perinatal and medical disorders, temperamental

difficultness in the child, psychological (over-) responsiveness of the parents,

parental insecurity and stress, psychosocial problems, and development of

vicious circles (19-20,32,61,82,89) have been documented.

The effects of insufficient and disturbed sleep in children include daytime

behavioural problems. The children do not necessarily indicate their sleep loss

by obvious daytime somnolence (119). On the contrary, a hyperactive

15

behaviour might be the only indicator of sleep deprivation in a child, with

resolution of the behaviour problem following successful treatment of the

sleep disturbance (46). The effect of chronically disturbed sleep on the parents

is extreme tiredness (30,47,98,125,129), and several studies have shown that

severe sleep problems in infants and toddlers (pre-school children) are

associated not only with extreme tiredness and but also with psychiatric (73)

and physical illness in the family, especially depression in the mother. The

parents of children with severe sleep problems often seek help, and

associations with marital problems (19-20,82,89) and even child abuse have

been described (30,61,67). A heightened risk of later development of

behavioural problems and school difficulties also exists for children with

severe sleep problems (31-32,67,83).

Treatment of sleep problems in childhood

The methods suggested in the management of sleep difficulties range from

total acceptance (117) (usually meaning taking the child to the parents´ bed)

to ignoring all crying, with many variations in between. A number of

treatment programs, based on behavioural techniques, have been described in

the literature (73). These include establishing night-time routines and rituals,

reorganizing settling and waking times, minimizing night-time attention by

gradually withdrawing reinforcers, controlled crying by graduated extinction

(progressive delay responding) or straight extinction ("crying it out"), and

scheduled anticipatory waking where parents arouse and then resettle their

child 15-60 minutes before expected spontaneous awakings.

Studies of these techniques show high rates of improvement (80-90 %) when

used with support from a therapist (5,63,97,101,118). Sedatives are

commonly used and may be helpful in the short-term, but have been shown to

have long-term benefits only when used in combination with a behavioural

16

programme (51). The studies carried out to investigate the success of

behavioural programmes rarely have follow-up periods longer than three

months (47,56,73,79,81,126,128).

Attention-deficit/hyperactivity disorder (ADHD) and

sleep

Sleep problems are frequently reported in children and adolescents with

attention-deficit/hyperactivity disorder (ADHD) (14,25,66,88,104,124).

Parents perceive children with ADHD to have greater sleep difficulty than

normally developing children, and anecdotal reports of practising clinicians

suggest that the sleep of children with ADHD is often reported by parents to

have been disturbed from the very beginning of the child´s life, ever since

infancy: "He has never slept through a whole night in his entire life."

Previously, based on clinical experience, sleep disturbance among children

with ADHD was so widely presumed that it was included as one of the

diagnostic criteria for the disorder (DSM-III; American Psychiatric

Association, 1980). However, further research and re-examinations (14) led to

the exclusion of the sleep disturbance criterion in the 1987 version of the

DSM-III-manual. The present diagnostic criteria for ADHD (DSM-IV, 1994)

are presented in Box 1.

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Box 1. Diagnostic criteria for attention-deficit/hyperactivity disorder (DSM-IV, 1994)*

A. Either (1) or (2):

(1) six (or more) of the following symptoms of inattention have persisted for at least6 months to a degree that is maladaptive and inconsistent with developmentallevel:

Inattention(a) often fails to give close attention to details or makes careless mistakes in

schoolwork, work, or other activities(b) often has difficulty sustaining attention in tasks or play activities(c) often does not seem to listen when spoken to directly(d) often does not follow through on instructions and fails to finish schoolwork,

chores, or duties in the workplace (not due to oppositional behaviour orfailure to understand instructions)

(e) often has difficulty organizing tasks and activities(f) often avoids, dislikes, or is reluctant to engage in tasks that require sustained

mental effort (such as schoolwork or homework)(g) often loses things necessary for tasks or activities (e.g., toys, school

assignments, pencils, books, or tools)(h) is often easily distracted by extraneous stimuli(i) is often forgetful in daily activities

(2) six (or more) of the following symptoms of hyperactivity-impulsivity havepersisted for at least 6 months to a degree that is maladaptive and inconsistentwith developmental level:

Hyperactivity(a) often fidgets with hands or feet or squirms in seat(b) often leaves seat in classroom or in other situations in which remaining

seated is expected(c) often runs about or climbs excessively in situations in which it is

inappropriate (in adolescents or adults, may be limited to subjective feelingsof restlessness)

(d) often has difficulty playing or engaging in leisure activities quietly(e) is often "on the go" or often acts as if "driven by a motor"(f) often talks excessively

Impulsivity(g) often blurts out answers before questions have been completed(h) often has difficulty awaiting turn(i) often interrupts or intrudes on others (e.g., butts into conversations or games)

A. Some hyperactive-impulsive or inattentive symptoms that caused impairment werepresent before age 7 years.

B. Some impairment from the symptoms is present in two or more settings (e.g., at school[or work] and at home).

18

C. There must be clear evidence of clinically significant impairment in social, academic,or occupational functioning.

D. The symptoms do not occur exclusively during the course of a PervasiveDevelopmental Disorder, Schizophrenia, or other Psychotic Disorder and are not betteraccounted for by another mental disorder (e.g., Mood Disorder, Anxiety Disorder,Dissociative Disorder, or a Personality Disorder).

* American Psychiatric Association. Diagnostic and statistical manual of mental disorders, 4th ed (DSM-IV). Washington, DC: American Psychiatric Press, 1994.

A detailed and systematic review of empirical research published since 1970

on sleep disturbances in children with ADHD, encompassing sixteen relevant

studies, was published in 1998 by Corkum, Tannock and Moldofsky (35).

They found that although subjective accounts of sleep disturbances in ADHD

were prevalent (based on sleep diaries, questionnaires and interviews),

objective verification of these disturbances (by polysomnographs, actigraphy

or video) was less robust (13). In fact, the only consistent objective findings

were that children with ADHD displayed more movements during sleep but

did not differ from normal controls in total sleep time (39,78,121). Further

studies by the same authors have confirmed this picture (34) and pointed to a

complex relationship between sleep problems and ADHD. In a recent study

(80) the conclusion was drawn that although subjective sleep difficulties are

common in ADHD youths, they are frequently accounted for by comorbidity

and pharmacotherapy.

However, when convincing evidence exists of sleeplessness in an overactive

child, attempts should be made to increase or improve the sleep because, in

some cases, the ADHD features may be the result of poor sleep (37,121). The

effects of sleep loss in children include tiredness, difficulties with focused

attention, low threshold to express negative affect (irritability and easy

frustration), and difficulty modulating impulses and emotions (37). In some

cases these symptoms may resemble ADHD. This has been reported in

children with obstructive sleep apnoea, with improvement of behaviour

following treatment (53).

19

ADHD-associated sleep disturbances may be improved by medication with

clonidine

(93,127). Improvements can also be achieved by administration of stimulant

drugs late in the day (29,93,121,127) even if insomnia secondary to stimulant

medication is a well-known adverse effect in some children (15,35,120).

Prospective, randomized, double-blind, placebo-controlled, long-term studies

of stimulant treatment for childhood attention-deficit problems have,

however, showed that for this patient group in general, early awakenings and

disturbed sleep tend to occur less frequently with stimulant treatment than at

baseline, i.e. without treatment (49).

In their review of the literature (35), Corkum, Tannock and Moldofsky

conclude that the exact nature of the sleep problems in children with ADHD

remains to be determined. Many relevant issues need new and better

methodological approaches. One interesting field is Periodic Limb

Movements in Sleep (PLMS) in children with ADHD, which has been

recently studied by Picchietti et al (90-91). PLMS are repetitive flexions of

the toes, feet, legs, thighs and/or the arms which recur periodically during

sleep. Sleep disruptions often accompany these movements, which in studies

using polysomnography and electromyography have been shown to be more

prevalent in children with ADHD than in control children.

Studies using retrospective data on sleep patterns in infancy among children

with ADHD are scarce. Trommer et al (124) compared 48 schoolchildren with

ADD (with and without hyperactivity) with 30 controls using retrospective

parental questionnaires which covered the ages 0 to 12 months and 1 to 4

years. The ADD children were reported to have had more frequent arousals

than the controls had both in the first year and from 1 to 4 years. However,

there is an obvious risk of selective memory bias in such studies.

20

Other prospective studies than the one included in this thesis on children with

severe sleep problems in infancy, concerning the risk of later development of

inattention and hyperactivity-impulsivity, have to our knowledge not yet been

performed.

21

Aims of the studies

The aims of this 5-year prospective study were:

1. to provide epidemiological data on infant sleep-wake characteristics and

the prevalence and severity of sleep problems in a general population, as

perceived by parents

(Study I),

2. to explore medical, psychological and psychosocial child and family

characteristics associated with severe infant sleep problems (Studies I och

II),

3. to investigate the changes in sleep in a group of severely sleep-disturbed

infants after a specific treatment programme combining behavioural

technique and interdisciplinary family work (Study III),

4. to prospectively follow and compare a group of children identified with

severe sleep problems in infancy with a control group regarding the

development of symptoms of attention-deficit/hyperactivity disorder

(ADHD) (Study IV) and regarding further sleep pattern (Studies III and

V),

5. to explore factors in infancy which are associated with subsequent

development of ADHD in later childhood (Study IV).

22

METHODS

Design and subject selection

Study I

A total community sample of 2 518 infants aged between 6 and 18 months

were approached using a parental questionnaire. The response rate was 83 %.

Data from the collection procedure point to a non-selective drop-out, not

reflecting presence or absence of sleep problems. Severe sleep problems as

defined by the ICSD were found in 129 of the children (6.2 %) who were

studied in detail, with the rest of the population as controls.

Studies II-IV

Those 6 to 12 month-old children in the community sample who fulfilled

specific inclusion criteria for severe and chronic sleep problems (N = 27)

created the case group.

Inclusion criteria were:

- the child should be reported by parents to have a prolonged sleep latency

and over three nightly wakings occurring at least five nights per week. This

pattern of disrupted sleep should have prevailed for a period of at least

6 months.

- infant age between 6 and 12 months at inclusion in the study. (The reason

for restriction of the study to this age group was limited resources and the

ambition to investigate all children with severe sleep problems in a specific

population with home visits by the same paediatrician).

23

- full-term pregnancy with a birth weight of greater than 2 500 grams and

gestational age of at least 37 weeks.

- the parents should report the need of help. (This criteria turned out to be

unnecessary. All parents of children with the above-mentioned sleep

problems reported themselves to be in need of help. This was true even for

the non-European parents in this group, who co-slept with their breastfed

child according to their cultural norms. They all reported much need for

help with their child´s sleep within the context of the Swedish

industrialized society).

- the parents should be able to communicate in Swedish or English.

The exclusion criterion was:

- signs of significant organic causes of the sleep problem identified by

hospital examinations and laboratory tests.

Among the 28 families with infants fulfilling the set inclusion criteria,

(i.e. 2 % of the sample of 1 259 children aged between 6 and 12 months), one

family was not able to participate because they moved to another part of the

country. The other 27 families agreed to participate in the study. Sixteen of

the children were boys, eleven girls. None of them fulfilled the exclusion

criteria. All were basically healthy and went further in the study.

A control group was matched with regard to age and sex. The infant of a

particular sex who fell next in age sequence to the case infant in the Child

Health Center (CHC) register was chosen as a control. A further criterion was

that no sleep problems were reported on the questionnaire. Of 33 families

contacted, 27 agreed to participate.

24

Study V

In addition to the original control group, an extended control group was

created from the questionnaire – study I, consisting of four control children

for each case child. The controls were matched for age and sex, had no

reported sleep problems in the questionnaire at 6 to 12 months and were still

living in Sweden five years later.

Procedure and measures

Study 1

In January 1996 data were collected in a parent population study in the urban

district of Uppsala (the fourth largest municipality in Sweden). A

questionnaire, constructed especially for this study, was sent to all parents

with an infant born during the period from July 1994 to July 1995.

Questionnaire on sleep and background factors

The instrument covered the following areas: social and demographic

background, pregnancy and birth, sleep-wake characteristics, parental

behaviour, and feeding problems.

Most items in the questionnaire had a 5-step response scale with verbal

definitions of each scale step. A few items had response categories with

yes/no answers. The items were constructed in close accordance with the

ICSD classification in addition to subjective parental rating scales of the

severity of the problem. No validation process was possible for these scales,

since the explicit aim of the questions was to measure early sleep problems as

perceived by parents.

25

Study II, III, V

A home visit was scheduled when parents had received information about the

study and agreed to participate in the research project.

The author, who is a clinically active a paediatrician, interviewed the parents

about the infant’s sleep characteristics and family members’ previous and

present state of health. The infant’s interaction with the parents and normal

activity were observed, and the development of the child was assessed. A

thorough somatic examination of the child was also performed. Information

about demographic variables, perceptions of parenting and infant

temperament, life events and social support was collected in a questionnaire

that the mothers filled in after the home visit. The following week the parents

kept a special Sleep Diary of the sleep of their child. This diary was brought

to the special CHC center where the parents of the children in the case group

received advice, counselling and support in the form of a behavioural training

programme, in order to overcome the sleep problems of their child. The sleep

programme was carried out as detailed later. The paediatrician was of course

aware of the group status of the families visited. In order to reduce the

possible influence of this, the assessments of the psychosocial problems were

compared with data from the CHC and the medical records of all the children.

This was done by two independent assistants who were blind to infant group

status.

One month after inclusion in the study the parents in the problem group were

sent another 7-day sleep diary and a general questionnaire about their opinion

of the help received. At one year, two and a half years and five years,

respectively, from inclusion, contact was made by mail, and a questionnaire

and sleep diary were filled in by the parents of both the problem group and

the control group. For ethical reasons the study design did not include any

placebo intervention group (i.e. untreated children in the problem group), as

26

studies of behavioural programmes have shown high rates of improvement

(80-90 %) (5,63,97,101,118).

Questionnaires on sleep, family and infant characteristics, and

sleep diaries

The home visit covered questions about sleeping characteristics and the state

of health of the family and comprised open-ended questions, scored on

yes/no, 3-step or 5-step response scales. Sleep characteristics were covered in

five questions that allowed a classification of the child’s sleep

pattern/problem according to the definitions of ICSD, 1990, in a 24-hour

recall of the infant’s sleep, and questions about the circumstances surrounding

getting the child to sleep. The following week the parents filled in a special

Sleep Diary of the sleep of their child in which the amount of night and

daytime sleep was recorded. In addition information was recorded about clock

time into bed, sleep latency, settling time, arousals (time and length), morning

wake time, in bed awake – and out of bed time, and day time naps (time and

length). All data were measured in minutes. Parental and sibling sleep

problems during infancy were also probed. In order to assess the infant’s

general development, the procedure used at the CHC was employed (National

Board of Health and Welfare, 1991), including an assessment of gross and

fine motor skills, as well as language, social, cognitive, and neurological

development. Development was rated on 5-step scales ranging from very late

for age to well above chronological age level.

The health status of the family was addressed by two questions about medical

pregnancy and delivery complications, and by four concerning the family’s

contact with medical care and health status during the infant’s lifetime.

Answers to questions about health were supplemented with information from

medical records.

27

Assessment of the psychosocial situation of the family was based on self-

disclosure during the medical interview (e.g. reports of unemployment,

financial restraints, conflicts between the infant’s parents, conflicts with the

infant’s grandparents, family health problems, substance abuse). Psychosocial

problems were rated by the author as none, minor or major. In order to reduce

the possible influence of the authors´ knowledge of the group status of the

child, the assessments of psychosocial problems were compared with data

from the CHC and medical records. This was done by two independent

assistants who were blind to infant group status. The findings showed an

interrater correlation of r = 0.95 when one assistant’s ratings of the listed

observations of the paediatrician (author) was compared with the other

assistant’s ratings of data from the CHC and medical records. The interrater

correlation between the author’s ratings and the assistant’s ratings was

r = 0.98.

Information about family demographic variables (ethnicity, age and

education, employment, housing and financial situation, and number of

children) was gathered by a questionnaire mothers completed after the home

visit. Maternal perceptions of parenting were obtained through semi-

structured questions scored by mothers on 5-step response scales. The

inventory, a revised Swedish version of R. Abidin´s Parenting Stress Index

(PSI; Abidin, 1990) (2,74,130), contained 34 items in 5 subscales that focused

on parental feelings of competence/incompetence, restriction by parental role,

social isolation, spouse relationship problems, and health problems.

To measure infant temperament, the Baby Behaviour Questionnaire (BBQ)

and the Toddler Behaviour Questionnaire (TBQ) (54-55) were used. In the

methodological work with the TBQ, a construct corresponding to difficultness

has been developed, consisting of low manageability as the most important

28

characteristic. This construct, based on 5-step response scales, was used in the

present study.

The occurrence of infants’ problematic behaviour during the previous month

was reported by mothers in the home interview. Questions in the interview

were related to sleeping habits, thumb or dummy sucking, screaming and

whining, shyness, activity, restlessness and lack of concentration, temper

tantrums and the mothers’ perception of the child as troublesome, difficult to

handle. Maternal ratings on 2-point response scales (yes, no) were used.

Treatment programme

The methods used were based on the following premise which was explained

to parents: "During the night all children have some spells of light sleep

during which they easily wake. The problem is not that the child wakes up but

that it cannot slip back to sleep without the help of a parent rocking or feeding

it. The way the baby is settled to sleep in the evening will be its preferred way

of returning to sleep after natural wakings. In order to have a child that returns

to sleep by itself, you have to accustom it to settle on its own".

The programme included:

Controlled crying

Controlled crying, or more positively called controlled comforting, implies

that the parent spends a few minutes settling the child, then leaves it for

gradually increasing lengths of time with brief and warm, but firm,

reassurance and resettling in between, yet avoiding taking the child out of its

cot. No feeding is given, and the child should be drowsy but still awake when

put into bed. The intervals between reassurance increase from initially one

minute up to five minutes at most. This procedure was used (if needed) every

29

time the baby was settled by day or night, and whenever it woke and cried at

night. Consistency was emphasized. A nurse and a child psychologist

supported the parents by daily telephone calls for 1-2 weeks and then tried to

help them to develop confidence in the programme and to manage on their

own.

Day and night routines

Overtired babies do not sleep well at night. Two daytime sleeps of about 1-1½

hours were encouraged, together with a regular pattern of feeds, play times

(with plenty of parental involvement) and sleep. The precise times could be

varied somewhat to fit in with each family situation, but it was important to

ensure that feeds and sleep were separated.

General help and information were given as appropriate, e.g. about nutrition,

feeding practices and play techniques. The great range of normal behaviour

and development was discussed with the parents.

Parents were encouraged to be agreed about the programme management and

to find ways to get support and time out for themselves in order to maintain

the energy needed to care for a baby or toddler. Problems such as relationship

stresses, social problems, depressive moods and parental health problems

were addressed if appropriate.

Medication

Five children with deeply depressed and exhausted parents were given

sedatives (alimemazine) for two weeks at the beginning of the programme, in

combination with the behavioural programme, in order for the parents to be

able to sleep directly and thereby regain some mental strength.

30

Study IV

A screening procedure for ADHD was carried out 4.5 to 5 years after

inclusion in the study, when the children had reached the age of 5.5 years. In

case of screen posivity, further assessment methods were used to diagnose

ADHD.

Screening instrument for ADHD

The screening procedures were identical for all children in the study and

comprised the following:

(i) a Parent Psychomotor Questionnaire (PPQ) containing four questions

pertaining to the child’s psychomotor development for completion by parents

and rated on a scale of normal/abnormal (95).

(ii) a previously validated Preschool Questionnaire (PSQ) comprising six

items pertaining to the child’s attentional, motor, language and perceptual-

conceptual capacity for completion by the preschool teacher and rated on a

scale of normal/abnormal (96).

(iii) a motor examination, performed by a nurse who had received special

training. The examination was carried out at the CHC, according to a

standardized screening of motor abilities that comprised six items rated on a

scale of normal/abnormal (48).

The screening procedures were in accordance with the battery recommended

by the Swedish National Board of Health and Welfare for screening for minor

neurodevelopmental/psychiatric disorders in preschool children at the CHCs

(72,96).

31

The criteria for screen positivity were: (a) one or more abnormal scores on

either the PPQ or the PSQ in combination with one or two abnormalities on

the motor examination or (b) three abnormalities on the motor examination.

Children not fulfilling these criteria in the screening procedure were

considered screen-negative. The criteria for screen-positivity were chosen so

as to identify children with ADHD as well as those with other

neurodevelopmental and neuropsychiatric problems (72).

The screening methods used in this study have been psychometrically

examined in previous Swedish studies (45,48,50,96) and have been found to

have good inter-rater and test-retest reliability.

Assessment methods

The assessment methods used for diagnosis comprised a detailed history,

psychiatric and neurodevelopmental examination, neuropsychological

assessment and speech/language evaluation.

The first assessment was performed by the local multidisciplinary district

CHC teams including the local psychologists, speech therapists,

physiotherapists and paediatricians outside the original sleep study

organization. Those teams were thus unaware of the group status of the

children examined. The psychological examination included an assessment of

development with Griffiths´ Developmental Scale II (52) in addition to a

detailed history and a clinical neuropsychological assessment of symptoms of

inattention, hyperactivity, impulsivity and behavioural problems. The speech

therapists used clinically accepted assessment methods of language

comprehension, phonological and grammatical problems, and the

physiotherapists administered the Scandinavian motor-perceptual scale MPU

32

(57). The local paediatrician performed a paediatric physical examination of

the child with the focus on neurological functioning.

The second examining paediatrician with special training in child

neuropsychiatry (author M.T.) was unaware of the results of the previous

assessment performed by the local team members while performing the

second step in the assessment procedure, a home visit.

A medical, developmental and behavioural history was taken at a home

interview with the parents, using a standardized interview schedule. The

criteria for ADHD according to the DSM-IV were checked during this

interview, and each criterion was rated as 'definitively met', 'possibly met' or

'not met'. Inter-rater reliability of results obtained at this type of ADHD

interview is excellent (72).

Routine paediatric psychical examination and a brief neurodevelopmental

examination in accordance with the method outlined by Gillberg et al. (48)

were also performed at the home visit. Inter-rater reliability for this

standardised assessment is good to excellent with values of Pearson r ranging

from 0.68 to 1.00 for individual items (50).

Diagnosis

A comprehensive diagnosis was made in each case after all neuropsychiatric

assessments had been completed on the basis of the available information.

The diagnosis ADHD was set in children meeting the criteria for this

diagnosis of the DSM-IV (Box 1).

33

Statistics

For analysis of the data, the statistical program SAS (113) was used in all

studies. Group comparisons for dichotomous variables were executed with

Chi-square analyses or Fisher’s exact test (two-tailed) when an expected cell

value was less than 5. For variables with 3-,5- or 7-step response scales,

comparisons were made with t-tests. In group comparisons, the more

conservative alpha level of 0.01 was used because of the large number of

tests.

RESULTS

Prevalence and classification of sleep problems (Study I)Sleep problems: Descriptive dataPrevious sleep problems, irrespective of duration or type, were reported for

48.3 % of the total child population. At the time of inquiry, 40.8 % of the

children were reported to have episodes of evening sleep refusal, including

minor and transient problems. The sleep refusal was considered a severe or

very severe problem by 3.8 % of the parents.

With respect to night-waking, 62.3 % of the children had current problems,

irrespective of severity and duration. The night-wakings were considered a

severe or very severe problem by 5.6 % of the families.

Table 2 gives descriptive data for sleep refusal and night-waking problems

measured with parental rating scales. Table 1 gives descriptive data classified

according to the operational definitions of ICSD. The frequency of severe

sleep refusal and severe night-waking problems in the total child population

varied between 3.8 % and 6.2 % in these measurements, with the parents

34

being slightly more tolerant in their judgement and opinions than the ICSD

scales.

Table 1. Percentages of infants that fulfilled the ICSD criteria of severe sleep refusal

(limit-setting sleep disordera) and severe nightwaking problems (sleep-onset association

disorderb).

_________________________________________________________________________

Ages 6-12 months, N = 1048 Ages 13-20 months, N = 1018

Totc Chronicd Totc Chronicd

_________________________________________________________________________

Severe sleep refusal 4.9 3.2 3.1 2.2

Severe night waking

problems 8.6 2.7 3.8 2.6

_________________________________________________________________________

Note: More than one problem can be reported for a child.

a) The ICSD criteria of severe limit-setting sleep disorder require five or more episodes per night of

stalling, calling out or leaving the bedroom.

b) The ICSD criteria of severe sleep-onset association disorder require a prolonged sleep latency and over

three nightly wakings, or two or three wakings each lasting over 10 minutes or one lasting over 15

minutes. The awakenings occur at least five nights per week.

c) Percentages of children for which problems were reported.

d) Proportion of children with chronic problems.

Chronic sleep refusal (limit-setting sleep disorder) requires a duration of 3 months or longer. Chronic

night-waking problems (sleep-onset association disorder) require a duration of 6 months or longer.

35

Table 2. Percentages of infants for which current sleep problems were reported by

parents.

N = 2066.

Sleep refusal

no problem 59.2

minor problem 24.6

moderate problem 12.4

severe problem 2.6

very severe problem 1.2

Night waking

no problem 37.7

minor problem 32.3

moderate problem 24.4

severe problem 3.4

very severe problem 2.2

36

Family and infant characteristics associated with severeinfant sleep problems (studies I and II)

Comparison of severe sleep problem group (N = 129) with a

control group (N = 1937) (Study I).

Sex, siblings, age distribution, pregnancy and delivery

There were more boys in the problem group (60 %) compared with controls

(51 %)

(X2 (1, N=2066) = 4.01, p < 0.05). No significant difference was found for the

presence of siblings. The age distribution of children in the problem group

was uneven, with an overrepresentation of children aged 8, 9 and 10 months

(42%) compared with controls (26%)(X2 (1, N=2066) =15.09, p <0.001).

There were no significant group differences in prematurity or birthweight.

Family sleep problems

No significant group differences were found concerning previous childhood

sleep problems within the family (mother, father and siblings analyzed

separately). In the problem group 12 % of the mothers, 10 % of the fathers

and 27 % of the siblings were reported to have had sleep problems in the first

years of life. Corresponding figures among controls were 10.8 and 23 %,

respectively.

Parental behaviour

Significant group differences were found concerning kinds of parental

intervention when the child refused to go to bed in the evening and when the

child needed to go back to sleep after waking up in the night. In the problem

37

group the parents to a significantly lesser degree left the child alone to settle

by itself in the evening (8 %) compared with controls (16 %). The parents in

the problem group more often sang to their children, pulled the child in a baby

carriage, or preferably breastfed the child or offered it a bottle to settle with in

cases of sleep refusal in the evening, as shown in Table 3.

Table 3. Percentages of families who used different kinds of parental intervention to get

the child to sleep in situations when this was problematic.

________________________________________________________________________

Problem group Control group X2

N = 129 N = 1 937

In the evening

Leaves the child to settle alone 8 16 6.32*

Sings for the child 53 40 7.70**

Pulls the child in a baby carriage 8 3 9.92**

Breastfeeds the child / gives a bottle 63 38 31.13***

In the night

Puts their hand on the child 39 26 10.90***

Rocks/holds the child in their

arms 36 23 11.59***

Sings for the child 23 12 14.89***

Takes the child to the parents´ bed 65 50 10.72**

Breastfeeds the child / gives a bottle 70 48 36.04***

_________________________________________________________________________

Note: Chi-square statistics are given from comparisons of the problem group with the control group with

df = 1 in all tests.

* p < 0.02

** p < 0.01

*** p < 0.001

38

During the night the parents in the problem group also intervened more than

the control group parents in situations when the child woke up and needed to

go back to sleep. Table 3 shows significant group differences in the following

interventions: putting parents’ hand on the child, rocking/holding the child in

parents’ arms, singing for the child, taking the child to the parents’ bed and,

above all, breastfeeding the child or giving the child a bottle.

Feeding problems

Feeding problems were defined here as difficulties in eating solids, with

refusal and preference for breast-milk or formula. Comparisons of the group

with severe sleep problems and the control group revealed significant

differences. Feeding problems were reported for 33 % of the problem group

compared with 15 % of the controls

(X2 (1, N = 2 066) = 29.33, p < 0.001), and 18 % of the problem group

parents reported themselves as being in current need of help with the feeding

problem of their child, compared with 6 % of the control parents (X2 (1, N =

2 066) = 25.03, p < 0.001).

Health

Comparisons of the presence of eczema, allergy and nutritional intolerance

between the problem group and the control group showed no significant

differences. Neither could any differences be found concerning frequency of

occurrence of diarrhoea, constipation, colds, otitis or fever. However, more

worries and anxiety concerning infant health were reported in the problem

group than in the control group. Although there was no difference in the

frequency of colds or ear infections between the groups, the parents of the

affected children in the problem group were significantly more worried over

the diseases than were corresponding control parents: colds t (df=125) = 3.37,

p < 0.001; otitis t (df = 551) = 3.69, p < 0.001.

39

In this study severe sleep problems were found to be correlated with parental

worries and anxiety concerning infant health (although the children were

reported as being as healthy as the controls), infant feeding problems and

intensive parental interventional behaviour (especially feeding) during the

evening and night. A common factor of insecurity in the parental role is

suggested.

Comparison of severe and chronic infant sleep problem group (N

= 27) with a control group (N = 27) (Study II)

Sleeping characteristics

More case infants were breastfed (59 %) than were controls (15 %) (X2 (1, N

= 54) = 11.44, p < 0.001). Most case infants (78 %) were fed immediately

before going to sleep, whereas this was true for only 37 % of the controls (X2

(1, N = 54) = 9.16, p < 0.01).

Feeding was used by the parents of case children as a method of getting the

child to sleep. Consequently most case infants (18 out of 27,67 %) fell asleep

in the arms of their mother, whereas most of the control children fell asleep

alone in their own beds (16 out of 27,59 %). These differences, however,

were not significant.

During the night most of the case infants (56 %) slept in their parents’ bed

(co-sleeping), whereas this was very uncommon among controls. Only one of

the control children slept in the parents’ bed; the remaining control children,

26 out of 27 (96 %), slept in their own bed (X2, (1, N = 54) = 17.41, p <

0.001). Frequent night meals (5 meals or more) were given to 44 % of the

case infants. This pattern was not seen in any of the control children. The

majority of the controls (63 %) was not fed at all during the night.

40

Evaluation of sleep data (except that polysomnographic monitoring was not

used in this study) showed that 22 of 27 children in the problem group had a

sleep pattern consistent with the diagnosis, nocturnal eating (drinking)

syndrome. Four children in the problem group fulfilled criteria of sleep-onset

association disorder, and one child exhibited a sleep pattern characteristic of

schedule disorder.

Significant group differences were found concerning the childrens’ sleeping

time during the night based on a 7-day parental report in the form of a

detailed Sleep Diary (Table 4).

The case children’s mean sleeping time during the night was one and a half

hours less than that of the controls, and the case infant group woke up

significantly more often. A tendency towards group difference was also seen

concerning the children’s sleeping time during the day, with the case children

taking shorter day time naps. Over a 24-hour period the mean sleeping time

for the case children group was two hours less than that of the control group.

Table 4. Results from analyses of variance with means and standard deviations for

sleeping characteristics.

Cases (N = 27) Controls (N = 27)

M Sd M Sd p value*

Night-time sleep (in minutes) 557.7 78.78 647.6 45.36 0.0001

Number of night wakings 6.0 3.01 0.7 0.78 0.0001

Night time awake(length of arousal time atnight in minutes) 71.8 37.39 8.5 9.73 0.0001

Daytime sleep (in minutes) 107.6 61.11 138.1 43.60 0.0400

Sleep latency at bedtime 30.0 32.67 10.6 6.85 0.0053

Total sleep during a 24-hourperiod 665.3 91.54 785.7 47.42 0.0001

* p value from t-tests

41

The clock time into bed did not differ between the case and the control group,

but the sleep latency period was longer and, consequently, the settling time

later in the case group compared with the controls. No significant group

difference was found concerning morning wake time.

Previous sleeping problems in the family were more common among case

families (67 %) than among controls (7%) (X2 (1, N = 54) = 20.33,

p < 0.001).

Demographic characteristics

All mothers, except three in the case group and one in the control group, lived

with the

infant’s father. A significant difference was found with respect to parents’

ethnic origin

(X2 (1, N = 54) = 7.48, p <0.01); a higher proportion of case infants had a

non-Swedish parent. Even more uneven was the proportion of non-European

parents in the groups; 37 %

of the case children had one (7 %) or both (30 %) parents who originated

from a non-European country (Africa, South-America, Asia), whereas this

was true for only 4 % of the controls (X2 (1, N = 54) = 9.24, p < 0.01).

During the same time as this study was made, data were presented from the

Swedish Central Bureau of Statistics (SCB), in which Uppsala, where our

study was performed, was divided into areas of high, medium, and low-

income structure, based on family income. The majority of the case children

lived in low income areas, and a significant group difference was found

concerning family income, with financial problems reported in 48 % of the

case families compared to 11 % of the controls (X2 (1, N = 54) = 8.88,

42

p <0.01). Regarding housing conditions, the majority, 81 %, of the children

with sleep problems lived in blocks of flats as did the majority of the controls,

59 %. However, 37 % of the control children lived in a detached house in

residential districts compared with 11 % of the cases. This difference in

housing conditions was, however, not significant.

No significant differences between cases and controls were obtained for the

other demographic characteristics.

Health characteristics

A significant group difference was found concerning the psychosocial

situation in the family; case families had more psychosocial problems than the

control group. Comparisons of other variables related to somatic health in the

family and of infant development revealed no significant findings. Chi-square

analyses showed no significant differences in pregnancy and delivery

complications.

The parents of the case children reported symptoms related to chronic loss of

sleep with extreme tiredness and lack of energy (81 %). Feelings of

hopelessness, restlessness, inability to relax and irritation were common.

Many parents also reported sadness and crying and a sense of being close to

mental breakdown. Somatic and psychosomatic complaints were very

frequent (59 %) among parents of case children, such as headaches, pains in

shoulders and back or "the whole body", vertigo and dizziness, heart-

palpitation, fainting fits and loss of weight. Sexual problems were common

(33 %).

The parents of case children suffered from exhaustion, both physically and

mentally, and 11 case mothers (41 %) fulfilled the ICD-10-criteria for a

severe depressive episode. None of them had sought psychiatric help. Fifty-

43

two per cent of the case mothers reported feelings of guilt in relation to their

child, compared with 11 % of the controls

(X2 (1, N = 54) = 10.39, p <0.01). The pattern of interaction with the child

was affected, and 78 % of the case mothers reported feeling that the locus of

control of the situation and interaction was in their child. This feeling of being

controlled by the child was very uncommon in the control group, 11 % (X2 (1,

N = 54) = 24.30, p <0.001).

Although depressed and in expressed need of help, only 20 of the 27 case

parents (74 %) had asked for help at the Child Health Center. In the parent

questionnaire population study, the corresponding figure was 93 % among

parents who had experienced any sleep problem, including minor and

transient in their children. The reasons why the parents of the children with

the most serious sleep problems did not seek help were reported by them to be

difficulties with language, lack of energy and a sense of guilt that the sleep

problem of their child was their own fault.

Perceptions of parenting

Maternal perceptions of parenting differed significantly between groups.

Mothers of case infants had less positive perceptions of parenting and

experienced more parental stress, in particular feelings of incompetence and

restriction by the parental role, as well as perceived health problems.

Infant difficultness and problematic behaviours

Comparisons between case and control infants regarding difficultness and

problematic behaviours showed a significant group difference for

temperamental difficultness. Regarding problematic behaviours, a significant

group effect was found; cases had more problems than the control group. In

44

the assessment of the development of the children the paediatrician also rated

their activity level. More case infants were rated as very active (30 %)

compared with controls (0 %) (X2 (1, N = 54) = 9.39, p <0.01).

Independent ratings by the mothers revealed the same group difference; more

case infants were rated as very active (52 %) than were controls (0 %) (X2 (1,

N = 54) = 18.90, p <0.001.

Infants with severe and chronic sleep problems thus showed normal health

and general development, and had a normal growth pattern. Early severe sleep

problems were, however, found to be associated with a higher incidence of

breast-feeding, frequent

night meals as a method of putting the infant back to sleep, co-sleeping, and

previous sleeping problems in the family. They were also associated with

non-European origin, living in a low income area and with psychosocial

problems in the family, including financial problems, exhaustion and

depression in the mother, less positive perceptions of parenting and more

parental stress. There were also feelings of incompetence and restriction by

the parental role, as well as perceived health problems. An association with

infant difficultness, high activity and problematic behaviours was found.

Short-term and long-term outcome of an

interdisciplinary sleep treatment programme (Study III

and V)

Three sets of parents found that they could not agree with the premise on

which the programme was based, and therefore chose to leave the programme

without even trying it. Their children were reported to have unchanged severe

sleep problems one month later.

45

At the evaluation one month after the treatment programme had been initiated

in the 24 motivated families, significant changes had taken place. The average

number of times the babies woke up had decreased from 6.0 to 1.8 times per

night and night-time sleep had increased by an average of 67 minutes

(Table 5). The difference in total sleep during a 24-hour period between the

problem group and the control group had been reduced by 55 %, and the

difference in night-time sleep between the two groups had been reduced by

75 %. Ninety-two percent of the parents reported their children to have a good

sleep after treatment and were satisfied with advice and support. The sleep

pattern remained unchanged for two of the 24 problem children treated, and

no child had exaggerated sleep problems after treatment.

Table 5. Results from analyses of variance with means and standard deviations for

sleeping characteristics in the problem group, before and after treatment. N = 24.

Before treatment After treatment

M Sd M Sd p-value*

Night-time sleep (in minutes) 557.6 82.39 625.5 80.31 0.0059

Number of night wakings 6.0 3.17 1.8 1.48 0.0001

Night time awake (lengthof arousal time at night in minutes) 76.2 37.13 22.6 25.75 0.0001

Daytime sleep (in minutes) 102.5 60.66 106.1 39.44 NS

Total sleep during a 24-hourperiod 660.1 95.92 731.5 79.58 0.0074

*p value from t-tests

46

One year after the accomplishment of the sleep programme, the previous

problem group still had a good sleep. No significant differences were found

between the sleep duration of the formerly sleep-disturbed children and their

controls.

The follow-up after two and a half years showed no significant group

differences. The former problem group still slept as well as the controls.

Thus the changes were stable over time. The families in the case group also

managed to maintain the achieved changes in infant sleep behaviour on their

own; continuous therapist support was not necessary. This was true even for

formerly depressed and psychosocially burdened parents.

Five years after the initial contact the former problem group still slept as well

as the controls according to the parental ratings of sleeping characteristics

(Table 6). Concerning sleeping behaviour there was a tendency for the case

group to be judged by their parents to move more and be more restless during

sleep than were the controls. For the children in the problem group that at the

age of 5.5 years fulfilled the criteria for the diagnosis of attention-

deficit/hyperactivity disorder, this group difference in nocturnal restlessness

and movements reached significance (p < 0.0001). With respect to the

remaining sleeping behaviours as well as sleeping characteristics, the children

with ADHD as a group did not differ from the controls.

47

Table 6. Results from analyses of variance with means and standard deviations for

sleeping characteristics and behaviour during sleep, five years after initial contact

Cases (N = 24) Controls (N = 26)

M Sd M Sd p value*

Night-time sleep (in minutes) 628.8 56.08 639.5 43.79 NS

Number of night wakings 0.5 0.67 0.3 0.46 NS

Night time awake (length ofarousal time at night in minutes) 4.7 8.10 1.2 2.10 NS

Daytime sleep (in minutes) 0.0 0.00 0.0 0.00 NS**

Total sleep during a 24-hourperiod 628.8 56.08 639.5 43.79 NS

Behaviour (5-step scales): Cases (N = 24) Controls (N = 107)

Snoring or breathing

difficulties during sleep 0.3 0.45 0.5 0.92 NS

Head banging 0.2 0.65 0.0 0.23 NS

Restless movements 1.2 1.34 0.4 0.63 0.0123

Nightmares 0.7 0.65 0.5 0.50 NS

Sleep terrors (Pavor Nocturnus) 0.3 0.54 0.1 0.36 NS

Sleep walking (Somnambulism) 0.2 0.67 0.1 0.36 NS

Sleep talking (Somniloquy) 0.8 0.83 0.5 0.54 NS

Tooth grinding (Sleep Bruxism) 0.3 0.78 0.5 0.94 NS

Bedwetting 0.8 1.39 0.3 0.77 NS

Anxiety in the settling situation 0.1 0.29 0.1 0.41 NS

Protests against going to bed 1.0 1.41 0.5 0.71 NS

* p value from t-tests

** All values are the same for one class level

48

Factors associated with subsequent development of

attention-deficit/hyperactivity disorder (Study IV)

Of the 27 children with previously severe sleep problems in infancy (cases),

25 participated in the screening procedure for ADHD. Two families had

moved (returned to their home countries). Eight children in this group were

screen-positive and went further to assessment. Of those, one child did not

qualify for any diagnosis of neurodevelop-mental/neuropsychiatric disorder.

The rest, seven children in the former sleep problem group, met the criteria

for ADHD. Five of those children also had additional motor-perceptual

problems including gross motor, fine motor, perceptual and speech/language

dysfunction. Severe behavioural problems, dominated by conduct problems,

were seen in three of the children. One of the children was diagnosed by the

district paediatrician and the local CHC team before the screening study (at

4.5 years of age).

In the group of 27 control children, 25 agreed to participate in the general

screening. One family had moved and one refused to participate, referring to

lack of time.

One child among the controls was screen-positive but in the following

assessment procedure did not qualify for any diagnosis. To summarize, seven

of the former sleep problem children met the criteria for the diagnosis of

ADHD, compared with no child in the control group.

The diagnosis ADHD was thus significantly more prevalent in children who

previously had severe infant sleep problems than among the controls, 7/25

versus 0/25 (p < 0.01).

Factors associated with later development of ADHD in children with severe

infant sleep problems were sought by comparing infant data of those children

in the sleep problem group that later developed ADHD (N = 7) with infant

49

data from the rest of the problem group (N=20). Given severe sleep problems

in infancy, the following characteristics were associated with later diagnosis

of ADHD; psychosocial problems in the family, behavioural problems at

settling time (bedtime struggles) and a long sleep latency at bedtime (Table 7

and 8). There was also a tendency towards more behavioural problems during

daytime (problematic behaviours) in infancy, according to parental ratings

four and a half to five years earlier, in the children that later met the criteria

for ADHD. These children also tended to have been rated by the paediatrician

as very active as infants (Table 7).

50

Table 7. Means and standard deviations for health characteristics, infant difficultness,

problematic behaviours and sleeping characteristics at 6-12 months of age

Characteristic Children with ADHD Rest of former sleep problem group

(n=7) (n=20)

Mean SD Mean SD p-value*

Health characteristics

Infant health problems

Severe episodes 0.7 0.76 0.1 0.22 NS

Moderate 1.1 1.21 0.6 0.83 NS

Common, banal 2.0 1.00 1.5 0.83 NS

Infant’s contact with medical care Specialist, paediatrician 1.7 1.38 0.9 1.04 NS

General practitioner 1.9 1.46 1.1 1.10 NS

Psychosocial problems 1.0 0.00 0.5 0.61 0.0016

General development of the infant 3.3 0.76 3.2 0.37 NS

Activity level in infant 1.8 1.10 0.6 1.00 0.0296

Difficultiness 3.8 1.70 3.9 1.20 NS

Problematic behaviours 3.8 1.92 2.0 1.36 0.0234

Sleeping characteristics

Bedtime struggles (7-step scale) 6.6 0.55 4.9 1.39 0.0003

Sleep latency at bedtime (min) 55.4 46.6 21.1 21.25 0.0104

Night-time sleep (min) 567.9 116.14 554.2 64.68 NS

Number of night wakings 6.3 3.20 5.9 3.02 NS

Night-time awake (length of arousal time at night) (min) 76.7 47.26 70.1 34.58 NS

Daytime sleep (min) 116.1 85.72 104.6 52.46 NS

Total sleep during a 24-h period 684.0 137.53 658.8 72.94 NS * p value from t-tests

SD, standard deviation

51

Table 8. Number of families with different kinds of psychosocial problems when the child was 6-12 months old

Characteristic Children with ADHD Rest of former sleepproblem group

(n = 7) (n = 20)

Unemployment 2 0

Insecure employment 2 2

Single mother 2 1

Chronic disease/disablementin parent 2 2

Maternal depression 2 9

Serious family conflicts 2 4

Refugee status 3 2

Substance abuse 1 1

Previous assault andbattery against motherin the family 1 0

Father previously inprison 1 0 No psychosocial problem 0 11

Note: More than one problem can be reported per family

52

DISCUSSION

General discussion

Study I, performed on a large total community sample, confirms results from

other populations concerning the proportion of sleep problems as perceived

by parents

(20-22,67,69,92). The designation of infant sleep problems as an area of

major parental concern is obvious as parental reports showed that 16 % of the

infants aged between 6 and 18 months were judged to have moderate to

severe difficulties in falling asleep at night, and 30 % were reported to have

frequent night waking.

A core group of children with severe or even very severe sleep problems,

encompassing around 5 % of the population, could be identified in different

ways. About 4 % of the infants had sleep refusal problems that were

considered severe or very severe by parental rankings and by the more

operational definitions of ICSD. The corresponding figure for night-waking

problems was 6 %. Five per cent of the parents reported themselves as being

in current need of help.

Almost all parents had sought help at a CHC or more seldom at a paediatric

clinic. Only approximately half of the parents were satisfied with the help

obtained. Obviously something is lacking. Further studies of the interaction

between parents and professionals regarding sleep problems are needed in

order to determine the nature of the problem. Is there a problem in the

information provided, in the way it is provided, or are the parents’

expectations not in concordance with those of the profession? Further

research is urgent.

53

The delineation of factors associated with severe sleep problems was one of

the major aims of the study. Results both from study I and from study II

indicate that severe sleep problems are related to a number of factors. The

most striking factors associated with severe sleep problems in the parent

population study I were intensive parental interventional behaviour

(especially feeding) during the evening and night, parental worries and

anxiety concerning infant health (although their children were reported as

being as healthy as others) and infant feeding problems. The parents of the

children in the problem group seemed to be more insecure in their parental

role, a finding that was confirmed in study II. It is of course always difficult

to ascertain whether the behaviour of parents actually contributes to the

development of the sleep problem of the child or whether, because the child

acts in a particular way (will not settle or wakes and cries), the parents adopt

certain behaviours (eg rocking, feeding) to get the child to sleep. Works

undertaken by Kerr et al (68) in the area of prevention, however, support the

first view. Altering the parent’s behaviour in the early months of the child’s

life in a preventive study produced a statistically significant reduction in the

incidence of sleep problems in the intervention group when compared to a

control group at 9 months of age.

In study II, the closer study with in-depth interviews and examinations of the

sleep-disturbed infants, a number of factors associated with severe sleep

problems were found; characteristics of the child, of the parents and of their

psychological and social situation.

Infants with severe and chronic sleep problems showed normal health and

general development and had a normal growth pattern. Early severe sleep

problems were, however, found to be associated with a higher incidence of

breast-feeding, frequent night meals as a method of putting the infant back to

sleep, co-sleeping and previous sleeping problems in the family. They were

54

also associated with non-European origin, living in a low income area and

with psychosocial problems in the family, including financial problems,

exhaustion and depression in the mother, less positive perceptions of

parenting and more parental stress. There were also feelings of incompetence

and restriction by the parental role, and perceived health problems. An

association with infant difficultness, high activity and problematic behaviours

were found.

There are potential dependencies of certain of the demographic variables

(socio-economic status, ethnicity) and other variables (such as the rate of co-

sleeping, breastfeeding and nap frequency) which make it more difficult to

evaluate the contribution of each factor. However, we do not make any causal

conclusions but believe that it is of clinical importance for prevention and

early identification in child health care and clinical paediatric settings to be

able to point to and identify common patterns and risk conditions associated

with severe sleep problems.

Earlier findings were replicated in study II. More frequent night-waking has

been reported in breastfed infants, (3,9,26,40,71,89) and in children, most

often breastfed, who are given frequent nightmeals (3,89). An association

between co-sleeping all night and problems with bedtime struggles and night-

waking has also been reported by several studies (3,62,67,75-

77,86,98,114,125,129). Previous sleeping problems in the family have been

less investigated, and as an experienced researcher concluded: "little is known

about how variations in social expectations and norms are related to sleep

patterns or sleep problems" (100). A clear hereditary component is only

known in parasomniacs (64).

The association of sleep problems with psychosocial problems in the family,

including financial problems and overcrowding, is well known. (3)

Exhaustion (30) and depressive state, especially in the mother, have

previously been widely documented (73,76,98,129) as have less positive

55

perceptions of parenting (3,19,76). The parent-child interaction has been

studied, and oversensitivity to the signals of the child (20,89) as well as

insecurity in the parents have previously been found. Other contributors

include inadequate stimulation of the child, overanxiety, rigidity or

inconsistent handling from the parents (85) and health problems in the family

(76).

In recent years the temperament of the child has come into focus, and infant

temperamental difficultness in sleep problems has been documented,

(3,9,12,27,82,98-99) as well as problematic behaviour, (6,82,98,129). The

question of hyperactivity in the sleepless child is still controversial. It has

been stated that there is no convincing evidence that sleepless children in

general are hyperactive per se, and many of them "calm down" when their

parents change their attitudes and establish consistent routines (100). A core

group of true hyperactive children with sleep problems most certainly exists,

but in the majority of very active children, this activity level has been seen as

a consequence rather than a cause of their sleeplessness.

In study II we did not measure infant temperament before the sleep problem

started. This means that we cannot rule out the contribution of infants’

temperamental characteristics, and in study IV we later found that

approximately one in four children with severe sleep problems in infancy will

later qualify for the diagnosis of ADHD.

The results of study II are thus in line with earlier findings. What may be new

is the massive impact. Together, our results give a very complex, large-scale

picture of how the infant and family situation develops in cases of very severe

infant sleep problems. This is a picture in which multiple factors may amplify

each other in vicious circles. Characteristics of the child, of the parents and of

the situation may influence each other reciprocally. Of course there are

methodological limitations here. Interpretation of a multifactorial genesis of

56

the problem could not strictly be assigned from univariate analyses but a not

too improbable speculation is that when a vulnerable child with a

temperament characterized by difficultness, high activity, liveliness, a strong

will and stubbornness, and with heredity for difficulties in the regulation of

sleep and wakefulness, is born into a family with great psychosocial

problems, health difficulties or a tendency to depression in the parent(s), there

emerges a situation of excessive parental stress. Living in a foreign country,

with other child-rearing traditions and far away from relatives, as for the non-

European families (half of them refugees), adds even more stress to the

situation. The higher risk of sleep disturbances in children of first generation

immigrants, possibly due to a periodic effect of the stress of cultural change,

has recently been discussed by Rona et al (106). In such cases, the child’s

needs may exceed the parents’ capacity to meet them.

A feeling of insufficiency and insecurity in the parental role, desperation, and

finally depression, in the parent can be the result. The child, who probably

more than other children needs regularity and routines, is met instead by

resignation and easy solutions, such as pacifiers, on-demand breast-feeding

and co-sleeping. A chronic lack of sleep may be the result for the parents with

such consequences as extreme tiredness, physical and mental exhaustion,

irritability and relationship problems. For the child, there may be exaggerated

difficulties and behavioural problems that maintain the vicious circle.

Sleepless and depressed parents may have neither the energy to solve the

situation themselves nor an ability to grasp the situation and seek help.

Our study, with its very small drop-out rate, has identified an unselected

population of very difficult cases. Out of these, three quarters had

spontaneously sought help. The remaining quarter of the families had all

regularly visited their CHC for weight and length controls, and vaccinations,

but they had not mentioned the severe sleep problems of their child.

57

Thus, in order to discover the severe cases of sleep problems among infants

and young children, paediatricians and CHC need to actively ask parents

about the sleep of their children. Severe problems are otherwise

underreported.

In study III, infants with severe and chronic sleep problems showed

significant improvements in all night-time sleep characteristics after a

relatively short, but intensive and multi-disciplinary, intervention sleep

programme. The improvements proved to be stable over time, lasting the

whole follow-up period of five years. Continuous therapist support was not

necessary to maintain the changes in sleep behaviour. In fact no contact

between the special CHC team and the case families took place during the

follow-up period, besides the follow-up questionnaires and a few telephone

calls from three of the families.

Furthermore, earlier findings were replicated in this study. The benefits of a

brief behavioural intervention programme for improving sleep in infants and

young children have previously been documented (4,36,43,79,116,128). Of

course it cannot be ruled out that a gradual achievement of a normal sleep

pattern would have been the natural history for some of the children in the

problem group, even without treatment.

Controlled crying was only one part of the programme, but is perhaps the

most controversial one. It has been argued that leaving a child of this age to

cry for even short periods may affect its long-term feeling of security (117).

There is no support for this belief in our results. On the contrary, no case child

showed any signs of anxiety in the

settling situation one year after treatment, and the control children were more

prone to protest against going to bed than were the cases at the two-and-a-

58

half-year follow-up. However, further research on follow-ups of behavioural

sleep disturbances is needed.

Tendencies towards parasomnia (restless movements during sleep, head

banging, sleep walking) were seen in the case group at the follow-up at one

year but not at the two-and-a-half-year follow-up. After five years there was

only a slight tendency for the problem group to move more during sleep than

the controls. Further research will be necessary in order to determine whether

the infants with early sleep problems are more prone to develop true

parasomnia as they grow older than are the controls.

We believe the interdisciplinary approach in the clinical interventions to be

crucial (87,107). The whole family situation must be taken into consideration

(11) in order to overcome vicious circles of insecurity in the parental role,

feelings of insufficiency, desperation and, finally, depression. Given

understanding and the tools of a more secure way of parenting a child, the

majority of even heavily psychosocially burdened parents were able to

manage to help their child sleep well.

Validity of the results

The source of information is a crucial issue in the evaluation of sleep

disturbances in early childhood. The reliability and validity of parental reports

in the form of Sleep Diaries, the most common assessment instrument in

clinical practice, have been questioned in the literature. It has been stated that

parents might be unaware of many aspects of the sleep behaviour of their

infants during the night (6,108,111).

Consequently, a number of objective sleep measures have been employed,

including polysomnographic studies, pressure-sensitive mattresses, timelapse

video recordings and actigraphy (activity-based monitoring).

59

Results from studies comparing parental reports with objective sleep

measures (109) show that parents are acccurate reporters of sleep-schedule

measures such as sleep onset and sleep duration. They overestimate, however,

the time that their infants spend in actual sleep and underestimate the number

of their night wakings. According to that result, the short sleep duration and

many night wakings initially in our problem group should be regarded as a

minimum rather than be suspected of being an exaggeration by exhausted

parents. It could of course be argued that parents who co-sleep with their

children are likely to be aware of more night-time wakings than would parents

who sleep in different rooms. And since the parents in the problem group co-

slept with their children much more frequently than did parents in the control

group, one might suspect an artefact in reporting. However, the difference in

number of night wakings between the groups is very large, as is the difference

in length of reported time spent awake in the night. Even if an artefact cannot

be ruled out, it is not reasonable to believe it to have had a major impact on

the results.

The total community sample of 2 518 children, with a response rate of 83 %,

does not exhibit any known bias. Hence, results based on data from this

sample are assumed to be generalisable in children from similar

sociodemographic backgrounds, i.e. Scandinavian and probably North-

European larger cities.

Concerning reliability it has to be noted that strict alternative answers give

increased reliability but could be a threat to the validity by loss of

information. We tried to solve this dilemma by combining survey data with

interviews with both strict alternatives and open-ended questions.

60

The most controversial results in this thesis emerge in study IV, in which a

discussion of methodological issues is urgent. According to our results,

approximately one in four children with severe sleep problems in infancy will

later qualify for the diagnosis of ADHD. This is a figure which clearly

exceeds the estimated prevalence rate in this age group in a normal Swedish

population, 2.4-4 % (72). However, the small numbers of children involved in

the study, both at the screening and diagnostic stages, constitute an obvious

drawback. The small sample size limits the extent to which conclusions can

be drawn. We therefore regard our findings as preliminary and suggestive of a

need for larger-scale corroborative studies.

Nevertheless, even though numbers were small, the sample examined was

population-based and thus more likely to be representative of children with

severe infant sleep problems than most studies reporting on clinical case

series. Moreover, the drop-out rate at various stages of the study was very

small. The longitudinal prospective nature of the study is also an advantage.

Individual, family and social factors were associated with later development

of ADHD. Most striking were the associations with bedtime struggles and

psychosocial problems in the family. ADHD has been described as a

"biopsychosocial disorder", raising critical questions concerning the relations

between genetic, biological and environmental factors (122).

The findings that there were a number of family psychosocial problems

associated with the development of ADHD in infants with severe sleep

problems need a comment. It has been stated that parents of children with

ADHD have a high likelihood of ADHD themselves, and thus the family

dysfunction may be a by-product of the parents’ own adult type of ADHD-

related psychosocial difficulties.

61

What is then measured with the criteria of ADHD? Behaviour reflecting

genetic predisposition to a deviant functioning of the synapse–transmittor

system with difficulties in the regulation of sleep and wakefulness? Behaviour

reflecting psychosocial stress? Behaviour reflecting vulnerability and

difficultness in temperament? Potentially neuropathogenic events during the

pregnancy, perinatal and postnatal periods, which are known to be associated

with a heightened risk of development of ADHD (96), had been excluded

through the inclusion criteria. Again we return to the speculations in page 56.

And why do we see a positive development in sleep characteristics but not the

same concerning behaviour and development? This is an urgent task for

future research.

Conclusions and clinical implications

Sleep problems are a common parental concern during infancy, and many

parents seek advice and help from a CHC or paediatrician. Severe sleep

problems in infants and young children are complex problems with both

psychological and social factors involved, besides the strictly paediatric

issues. In addition, many families are not satisfied with the help they obtain

today. Even more serious, the severe cases are underreported by parents.

These findings have implications for the consultations at CHCs and for the

paediatric consultation in cases of severe sleep problems in infants and young

children. In order to discover the severe cases, health care personnel have to

actively ask parents about sleep problems. In addition to obtaining a thorough

history and making a complete medical examination of the child, physicians

also need to pay great attention to the situation of the parents, their worries,

and psychological and social conditions.

62

The possibility of a vicious circle developing deserves attention in clinical

interventions, which need to be interdisciplinary, taking the whole family

situation into consideration.

With a relatively short but intense treatment programme, combining

behavioural technique with family work, even severely sleep disturbed infants

and children, coming from families with depression and psychosocial

problems, can be helped to sleep well. Long-term improvements in sleep

pattern after the programme, extending over years, have been documented.

The results concerning the development of attention-deficit/hyperactivity

disorder call for heightened attentiveness and awareness among nurses and

doctors working with infants with sleep problems at CHCs and paediatric

clinics. Infant activity level, problematic behaviours and parent-infant

interaction have to be considered seriously, and both psychosocial,

behavioural and sleeping characteristics have to be taken into consideration.

Close follow-ups of children with combined severe and prolonged sleep,

activity and behavioural problems are recommended as neurodevelopmental

problems seem to be prevalent in this category of children.

63

ACKNOWLEDGEMENTS

The work reported in this thesis has been completed with the contribution of a

lot of individuals.

I wish to express my sincere gratitude to:

The parents and children who participated in the studies, for sharing their

experiences of life during the day and at night, and thus making this thesis

possible.

Professor Claes Sundelin, my supervisor, for excellent guidance in scientific

thinking, for generously sharing his knowledge and creating an ideal working

situation for me adapted to my needs, inspiring me and offering support and

advice from beginning to end in uncountable discussions.

Professor Jerker Hetta, my other supervisor, for expertise and advice in the

field of sleep research.

Colleages and friends at the Child Health Unit, Department of Women´s and

Children´s Health, Section for Pediatrics in Uppsala for comradeship and

generosity over the years.

Rut Magnusson for excellent secretarial assistance and personal support.

Monica Östberg, Margareta Dannaeus and Elisabet Hagelin, collegues at the

Special Child Health Center, for professional help in the sleep treatment

programme and Monica also in the work with Parental Stress Index, where

she generously shared her methods and data files with me.

64

Carina Lundin for patient and effective computer assistance.

Maurice Devenney for excellent English language service.

All nurses at the Child Health Centers (CHC) in Uppsala for their efforts and

important contributions to the work.

All paediatricians, psychologists, speech therapists and physiotherapists in the

local CHC teams for stimulating collaboration over the years.

My big family for loving care and support.

Financial support for this research was provided by grants from the Swedish

1st May Foundation and the Gillberg Foundation.

65

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