ARTICLE IN PRESS E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
12 (2008) 89 – 96
Official Journal of the European Paediatric Neurology Society
Original article
Parental perception of cold extremities and other accompanying symptoms in children with cerebral palsy Lena E Svedberga,b,, Erling Englundc, Hans Malkerc, Elisabet Stener-Victorina a
Institute of Neuroscience and Physiology, Sahlgrenska Academy, Go¨teborg University, Go¨teborg, Sweden ¨ rnsko¨ldsvik, Sweden Child and Youth Neurohabilitation, O¨rnsko¨ldsviks Hospital, SE-891 891, O c Department of Research and Development, Sundsvall, Sweden b
art i cle info
ab st rac t
Article history:
Cold extremities have been noted in non-walking children with cerebral damage compared
Received 16 March 2007
with healthy controls. Whether this is a general problem in children with cerebral palsy
Received in revised form
(CP) and associated with other symptoms is unknown. This study describes accompanying
12 June 2007
symptoms such as cold extremities, constipation, pain, sleeping disorders and impaired
Accepted 12 June 2007
well-being in children with CP as well as treatment the children have undergone. Associations between cold extremities and other symptoms borne by the children were
Keywords: Cerebral palsy cold extremities pain constipation sleeping disorders well-being
analysed and discussed. From information in postal surveys received from parents of children with CP, 107 children (60 boys and 47 girls) aged 5–13 years, mean 11 years 8 months (SD 2 years 11 months), were described and analysed. Besides neurological impairments, many children had cold extremities and pain, sleeping disorders, constipation, and impaired well-being. Most children had had one or more of these symptoms for over 1 year but the symptoms were largely untreated. Non-walkers generally had more symptoms than walkers. Although pain, constipation, and sleeping disorders may have different underlying causes in children with CP, these symptoms might also be mediated or aggravated by dysfunction in the autonomic nervous system. To improve the child’s wellbeing, early recognition and treatment of accompanying symptoms is important. & 2007 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.
1.
Introduction
Cerebral palsy (CP) is the most common cause of motor disability in children and had in period 1995–1998 a prevalence of 1.92 per 1000 live-born children in western Sweden.1 A revised definition of CP that spans other possible disturbances borne by the child 2 has been recently proposed. Further, several studies on children with CP have illustrated correlations between motor ‘‘severity’’ and other aspects of neurodevelopmental impair-
ment such as epilepsy, mental retardation, and sensory impairments.3–6 A sensory impairment like low skin temperature in the extremities has been noted in non-walking children with cerebral damage compared with healthy controls.7 Also, mean skin temperature was significantly lower at all foot measuring points in children with cerebral damage who were unable to walk compared with walking children with cerebral damage. Little emphasis has previously been accorded to peripheral skin temperature impairment in children with
Corresponding author. Child and Youth Neurohabilitation, O ¨ rnsko¨ldsviks Hospital, SE-891 891 O ¨ rnsko¨ldsvik, Sweden. Tel.: +46 660 894 19; fax: +46 660 894 79. E-mail address:
[email protected] (L.E. Svedberg). 1090-3798/$ - see front matter & 2007 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ejpn.2007.06.004
ARTICLE IN PRESS 90
E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
neurological disorders.8,9 The presence of thermal dysfunction has possibly been overshadowed by the child’s other disabilities. Whether cold extremities is a general problem in children with CP and associated with other symptoms is therefore unknown. But findings of unpleasant coldness in the extremities of stroke patients have been brought to the attention of the clinicians who care for these patients.10 It is therefore important to extend knowledge among those working with neurologically impaired children. The aims of this study were to describe these accompanying symptoms in a group of 5–13-year-old children with CP: cold extremities, constipation, pain, sleeping disorders, and impaired well-being; to elucidate whether cold extremities are a general problem; and to discern which accompanying symptoms are associated with cold extremities. Further, what treatment the children had been given was investigated. Associations between cold extremities and gross motor function and between cold extremities and other symptoms borne by the children were analysed and discussed.
2.
Subjects and methods
2.1.
Subjects
The study comprised 161 children with CP (born between 1989 and 1999) and enroled at 8 of 10 child and youth habilitation centres in the northern region of Sweden. Two centres were excluded since they had recently been involved in another survey organized by other researchers. A proxy report was used so that all children with CP in this age group, including those with intellectual impairment and insufficient communication skills, could participate in the study. Information about 110 of 161 children was received. Three children were excluded: one child had an inappropriate diagnosis, one child was born before 1989, and information on gross motor function was missing for one child. The final group comprised 107 children (67%), 60 boys and 47 girls, mean age 11 years 8 months (SD 2 years 11 months). Data on the missing children were unavailable due to professional confidentiality. The study was approved by the Ethics Committee at Umea˚ University, Umea˚, Sweden.
3.
Methods
The postal survey comprised 53 questions and took about 15 min to fill out. Ten questions were related to family, parental experiences, and services support and are not presented in this paper. The questionnaire included both open-ended and closed-ended questions and this part, focusing on the children, was designed to cover these topics: (1) gross motor functional ability, (2) accompanying neurological impairments, (3) general medical background, (4) the child’s condition and present treatment regarding skin temperature, pain, sleep, and constipation, (5) well-being, and (6) general health. Functional ability was classified with the Gross Motor Function Classification System (GMFCS). The response options in this question, which contained a parental report of
12 (2008) 89 – 96
GMFCS level relevant for the ages studied, was initially written in English (see www.npeu.ox.ac.uk/morris, Table 1). These options were translated into Swedish according to recommended guidelines 11 with independent forward and backward translations by bilingual translators. Discrepancies were discussed and resolved. GMFCS is a tool that classifies the functional ability of children with CP into five levels.12 The validity and reliability of the GMFCS were found to be good when professionals used it with children aged 2–12 years.13 Agreement between family reports and professional classifications has also been found to be excellent, which suggests that family reports of the GMFCS are a reliable method for measuring gross motor functional abilities of children aged 6–12 years.14 This is particularly useful in observational studies of larger populations when it is often not feasible for professionals to rate the children. Children and families at habilitation units are protected by professional confidentiality and may not be contacted directly for research purposes. So, the questionnaires were sent to the parents by their habilitation unit with an introduction letter and a prepaid envelope addressed to the unit. One reminder was sent to those who had not answered after 2 weeks. If the caregivers of a child were separated, both were sent the Table 1 – Criteria for five levels of Gross motor Function Classification System (GMFCS), relevant for ages studied GMFCS Level I Can walk on their own without using walking aids, and go up or down stairs without needing to hold the handrail and walks wherever they want to go (including uneven surfaces, slopes or in crowds) and can run and jump though their speed, balance and coordination may be slightly limited Level II Can walk on their own without using walking aids, but need to hold the handrail when going up or down stairs and often finds it difficult to walk on uneven surfaces, slopes or in crowds Level III Can stand on their own and only walks using a walking aid (such as a Kaye walker, rollator or any kind of crutches, walking sticks or canes etc.) and finds it difficult to climb stairs, or walk on uneven surfaces and may use a wheelchair when travelling for long distances or in crowds Level IV Can sit on their own but does not stand or walk without significant support and therefore relies mostly on wheelchairs at home, school and in the community and often needs extra body/trunk support to improve arm and hand function and may achieve self-mobility using a power wheelchair Level V Has difficulty sitting on their own and controlling their head and body posture in most position and has difficulty achieving any voluntary control of movement and needs specially supportive chair to sit comfortably and has to be lifted or hoisted by another person to move
ARTICLE IN PRESS E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
survey with a special introduction letter, which explained that both parents were being asked to participate in the survey. The answers in the questionnaire were unknown to the personnel at the habilitation unit; the return envelopes were forwarded unopened to the first author.
3.1.
were indicated by reporting for how many children the question had been answered. A limit of 2 SD for body mass index (BMI) analyses was chosen to detect over- and underweight children.15 Five answer options were given in the questionnaire to report presence and frequency of the accompanying specific symptoms. Four of these described a frequency range from ‘‘yes, every day’’ to ‘‘yes, a few times/ year’’ and the fifth option was ‘‘no never’’. The five options were divided into two groups, ‘‘yes’’ or ‘‘no’’ when the distribution of specific symptoms by gross motor function levels were analysed; and into three groups, ‘‘a few times/ week or more’’, a few times/month or less’’ and ‘‘no never’’, when the frequency of associated symptoms was described. Each of the five options was used when the correlation between cold extremities and other specific symptoms was calculated using the Spearman correlation. The Bonferroni method was used to adjust for multiple comparisons in the correlation analysis. The level of significance was set at po 0.05.
Statistics
Most of the statistics were interpreted descriptively using SPSS version 14.0 (SPSS Inc., Chicago, IL, USA). Three of the 107 children had two respondents each because their caregivers were separated. The parents of one of these three children answered the questionnaire together, so the answers were identical. For each of the other two children, the parental responses sometimes differed, and the answers from each parent were weighted by 0.5, which yielded decimal frequencies when the answers differed. Missing data Table 2 – Distribution of accompanying impairments and specific symptoms in walking and non-walking children with cerebral palsy Walkers
Impairments Learning disability Speech disorders Visual impairments Hearing impairment
Total
GMFCS I–III
Nonwalkers GMFCS IV–V
(n ¼ 64) n (%)
(n ¼ 43) n (%)
(n ¼ 107) n (%)
15 (23%)
25 (60%)
b
32 (74%)
47 (44%)
23.5 (37%)
36 (84%)
59.5 (56%)
3 ( 5%)
2 ( 5%)
b
5 ( 5%)d
Epilepsy Specific symptoms Cold extremities Pain
14 (22%)a
23 (54%)
37 (35%)d
36 (56%)
40 (93%)
76 (71%)
31 (48%)
34 (79%)
65 (61%)
Sleeping disorders Constipation Impaired wellbeing
23 (36%)
31 (72%)
54 (51%)
14 (22%) 31 (48%)
31 (72%) 26.5 (64%)c
45 (42%) 57.5 (55%)e
a
Pain
Results
4.1.
Gross motor function and muscle tonus
4.2.
Accompanying impairments and specific symptoms
Table 2 lists accompanying impairments and specific symptoms such as cold extremities, constipation, pain, sleeping disorders, and impaired well-being in walkers and nonwalkers. Fig. 1 illustrates the distribution of specific symptoms by GMFCS level. The higher the number of specific symptoms, the higher the proportion of non-walking children (Fig. 2).
n ¼ 63; bn ¼ 42; cn ¼ 41.5; dn ¼ 106; en ¼ 105.5.
Cold extremities
4.
The gross motor function was classified as GMFCS level I in 37% (40/107) of the children, as level II in 19% (20/107), as level III in 4% (4/107), as level IV in 21% (23/107), and as level V in 19% (20/107). The 107 children were divided into two gross motor functional groups: 60% (64/107) were classified as walkers (GMFCS I–III) and 40% (43/107) as non-walkers (GMFCS IV–V). Spasticity was reported in 77% (80.5/105) of the children and dystonia in 37% (37/99.5).
40 (38%)d
15 (23%)
91
1 2 (20 08) 89 – 9 6
Sleeping disorders
Constipation
Impaired well-being
Level I (n=40) Level II (n=20) Level III (n=4) Level IV (n=23) Level V (n=20) 0
50
100 0
50
100 0
50
100 0
50
100 0
50
100
% Fig. 1 – Distribution of accompanying specific symptoms by GMFCS level. The dashed line divides the children in walkers (above) and non-walkers (below).
ARTICLE IN PRESS 92
E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
40
40 Walkers
%
35
Non-Walkers
35
30
30
25
25
20
20
15
15
10
10
5
5 0
1
2
3 4 5 - - 0 1 2 3 Numbers of specific symptoms
4
5
Fig. 2 – Proportion of walkers and non-walkers by numbers of accompanying specific symptoms.
General medical background
Forty-five percent (48/107) of the children were born at full term (436 gestations weeks), 17% (18/107) between weeks 32 and 36, 15% (16/107) between weeks 28 and 31, and 17% (18/ 107) before week 28 while gestation period was unknown in 7% (7/107). Mean birth weight was 2.496 kg (range 0.422–5.150). At the time of the survey, mean reported weight for 81 children was 41 kg (range 15–125) and mean reported height for 84 children 1.44 m (range 1.08–1.83). BMI analyses of 81 children yielded that 22% (18/81) were underweight, 64% (52/ 81) normal weight, and 14% (11/81) overweight. Of 107 children, impairment in oral-motor function reflected by difficulties with food and liquid intake was present in 19% (20/107) and 16% (17/106), respectively. Severe problems, percutaneous endoscopic gastrostomy (PEG) feeding and gastro-oesophageal reflux, were reported in 11% (12/107) and 13% (14/106), respectively. These feeding problems occurred mainly in non-walking children. Incontinence occurred in 25% (26.5/107) of the children.
Pain was observed in 61% (65/107) of the children (Table 2) and 94% (61/65) of these had experienced pain for more than 1 year. The parental opinion was that pain, for example, was located in the muscles and joints or due to constipation. A larger proportion of non-walking children than of walking children was reported to have pain: 79% (34/43) compared with 48% (31/64), respectively (Table 2). Forty-two percent (18/ 43) of the non-walkers and 6% (4/64) of the walkers had pain a few times/week or more (Fig. 3).
4.6.
Sleeping disorders
Disturbed sleep such as delayed insomnia, disrupted sleep, early awake, or a combination of these was reported in 51% (54/107) of the children (Table 2), and 93% (50/54) of these children had had the disorder more than 1 year. Suggested causes of the child’s sleeping problems were difficulty to relax and calm down or presence of spasticity, anxiety, epilepsy, and pain. Sleeping disorders were reported more often in nonwalkers, 72% (31/43), than in walkers, 36% (23/64) (Table 2). Fifty-six percent (24/43) of the non-walkers and 20% (13/64) of the walkers had disturbed sleep a few times/week or more (Fig. 3).
4.7.
Constipation
Forty-two percent (45/107) of the children were reported to have constipation (Table 2), and all 45 had had the symptom more than 1 year. Parental opinions of possible causes for this problem were impaired motor ability, poor liquid intake, and intake of pureed food. More non-walking children, 72% (31/43), than walking children, 22% (14/64), had constipation (Table 2). Forty-two percent (18/43) of the non-walkers and 6% (4/64) of the walkers were reported to be constipated a few times/week or more (Fig. 3).
4.8. 4.4.
Pain
0
0
4.3.
4.5.
12 (2008) 89 – 96
Impaired well-being
Cold extremities
Parents reported that 71% (76/107) of the children had cold extremities (Table 2): 8% (6/76) only had cold hands, 24% (18/ 76) only cold feet, and 68% (52/76) cold hands and feet. Ninety-seven percent (74/76) of these children had had the symptom more than 1 year; 78% (59/76) had cold extremities at both normal and low room and outside temperatures and 22% (17/76) only at low outside temperatures. Forty-four percent (33/75) had told their parents about their cold extremities in words or by other means, and 56% (42/75) had not. Possible causes for cold extremities, in the opinion of the parents, were low blood circulation, impaired motor ability, and spasticity. Non-walkers, 93% (40/43), had cold extremities more often than walkers, 56% (36/64) (Table 2). Frequency of cold extremities a few times/week or more was reported in 77% (33/43) of the non-walkers and 44% (28/64) of the walkers (Fig. 3).
Well-being was thought to be impaired in 55% (57.5/105.5) of the children (Table 2). This had been observed for more than 1 year in 91% (50.5/55.5) of these children. Suggested causes were impairments and spasticity. Impaired well-being was reported for 64% (26.5/41.5) of the non-walkers and 48% (31/64) of the walkers (Table 2). The well-being was impaired a few times/week of more in 31% (13/41.5) of the non-walkers and in 13% (8/64) of the walkers (Fig. 3).
4.9. Association between cold extremities and other symptoms An association between cold extremities and sleeping disorders (r ¼ 0.43, po0.001), constipation (r ¼ 0.41, po0.001), pain (r ¼ 0.33, po0.01), and impaired well-being (r ¼ 0.32, po0.01) was observed. No association was found between cold extremities and spasticity, epilepsy, gestation age, birth
ARTICLE IN PRESS E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
Proportion of children
100 90 80 70 60 50 40 30 20 10 0 100 90 80 70 60 50 40 30 20 10 0 100 90 80 70 60 50 40 30 20 10 0
100 90 80 70 60 50 40 30 20 10 0
Cold extremities
Impaired well-being
Pain
100 90 80 70 60 50 40 30 20 10 0
Sleeping disorders
93
1 2 (20 08) 89 – 9 6
Constipation
r r ore less ve ore less ve r m h or o, ne r m h or o, ne o o k ont k ont N N ee ee s/w es/m s/w es/m e e tim tim tim tim ew few ew few A A Af Af
Walkers Non-walkers
r r ore less ve ore less ve r m h or o, ne r m h or o, ne o o k ont k ont N N ee ee s/w es/m s/w es/m e e tim tim tim tim ew few ew few A A Af Af Fig. 3 – Proportion of walkers and non-walkers by frequency of accompanying specific symptoms. weight, or BMI, even though the proportion of children with cold extremities who were underweight (27%; 16/59) was higher than the proportion of underweight children with no cold extremities (9%; 2/22). An association between cold extremities and sleeping disorders (r ¼ 0.45, po0.01) and pain (r ¼ 0.45, po0.01) was found in non-walkers; none was found in walkers.
Cold extremities (n=76) Pain (n=65)
n=31
Sleeping disorders (n=54)
n=29
Constipation (n=45)
4.10.
4.11.
General health
Parents estimated the general health of both walkers and non-walkers to be good or very good.
n=14
n=14 n=34 n=25 n=31
0 10 20 30 40 50 60 70 80 90 100 % Untreated children Treated children
Present treatment
No treatment was offered to 82% (62/76) of the children with cold extremities, 48% (31/65) of the children with pain, 54% (29/54) of the children with sleeping disorders, and 31% (14/45) of the children with constipation (Fig. 4). Similar distributions of treated and untreated children were found when the symptoms had been present more than 1 year. Initiative for treatment was generally taken by the family and not the professionals. Fig. 5 lists the different treatments for each symptom and their effect, according to the parents.
n=62
Fig. 4 – Proportion of untreated and treated children with accompanying specific symptoms.
5.
Discussion
Cold extremities were commonly noted in children with CP and seemed to be associated with other symptoms. Surprisingly, the children’s condition was largely untreated.
5.1.
Accompanying impairments and specific symptoms
The distribution of learning disability, hearing impairment, and epilepsy in the 107 children in this study was similar to
ARTICLE IN PRESS 94
E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
12 (2008) 89 – 96
Pain
Cold extremities Medication Oral support/advises Orthopedical technical aid Acupuncture Tactile stimulation TENS Bath/Heating pillow Extra warm clothes a Other help 0
5
10
15
20
25
0
5
Sleeping disorders
10
15
20
25
Constipation
Medication Oral support/advice Technical aid Physical training b Other help 0
5
10
15
20
Satisfied
25
0
5
10
15
20
25
Numbers of children
Not satisfied Missing Fig. 5 – Received treatments for accompanying specific symptoms and their effect, according to the parents. aBlanket or mattress, bMassage, relaxing music and occlusal splint for sleeping disorders; and massage and food rich of fibre for constipation. the distribution reported by Himmelmann et al. in 2006.6 Speech disorders were not assessed, and only severe visual impairment was recorded. Besides the accompanying impairments, a large proportion of the children with CP in our study were reported to have cold extremities, pain, sleeping disorders, constipation, and impaired well-being. Most of them had had one or several of these five symptoms for more than 1 year. In general, a higher frequency of symptoms was reported in non-walking children than walkers.
5.2. Association between cold extremities and other symptoms We found an association between cold extremities and pain, sleeping disorders, constipation, and impaired well-being. The question of why these symptoms are related remains. Low skin temperature may be caused by decreased peripheral blood circulation due to impaired motor ability. Pain could originate from muscles and joints or be a result of constipation and reflux. Difficulty to relax combined with spasticity, anxiety, epilepsy, or pain may affect sleep. Constipation could be linked to immobility, medication, poor liquid intake and use of pureed food. The child’s perception of these symptoms might result in impaired well-being and physical and mental disabilities in social situations. Another reason for low skin temperature might be altered autonomic control of vasomotor tone due to brain damage, as is thought to occur in adult stroke patients.10,16,17 Studies have also reported that disturbed sympathetic nerve activity may result in constipation 18 and pain 19 in adult patients. Surprisingly, little attention has been paid to the possibility of disturbed autonomic nervous activity in children with CP.7,8
Skin blood flow in the hands and feet is controlled by sympathetic fibres, 20 and the close relationship between pain and low skin temperature might be due to high tone in sympathetic vasoconstriction fibres.19 Pain has been reported to commonly occur in the daily life of children with CP and severe motor impairments compared to a control population.21 Chronic constipation is a common complaint in children with severe brain damage and was found to result from prolonged transit through the more proximal segments of the colon.22,23 Thus, impaired supraspinal coordinative influence on autonomic visceral nerves may result in intestinal dysfunction. Sleep is a complex neurological function. It has been suggested that the autonomic nervous system, which is involved in pineal melatonin secretion and sleep regulation, is affected in children with brain damage.24 So, cold extremities and the association with pain, constipation, and sleeping disorders could be linked to dysfunction of the autonomic nervous system in children with CP. The autonomic nervous system function in children with CP is largely unexplored. However, attempts have been done to investigate this area as several clinical symptoms in these children have been suggested to be a result from autonomic dysfunction. For example, 30 children with CP aged between 4 and 10 years were aged and sex matched with control subjects and power spectrum analysis of heart rate variability (HRV) was used to investigate the autonomic nervous system function.25 A disturbed balance in the relative activity of the sympathetic and parasympathetic nervous system was observed for children with CP. It was suggested that disturbed modulating effects on autonomic function due to brain lesion in children with CP might account for the presence of clinical
ARTICLE IN PRESS E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
dysautonomia such as bowel and bladder dysfunction, hyperhidrosis and poor cardiopulmonary endurance.25 Further, analysis of autonomic regulation using variational cardiointervalography (VCIG) has shown dysadaptation in the compensatory cardiovascular system in 157 children with CP.26 Severe adaptation was found in 73% of children with severe and very severe motor disorders and in only 32.8% with mild outcomes of CP.
5.3.
Treatment
Pain, sleeping disorders, constipation, and cold feet seemed to be accepted as inevitable consequences of the child’s neurological impairment with treatment delays of over 1 year for many of the children. Communication difficulties may compound the delay, because the child is often unable to express discomfort. Higher priority is probably given to other aspects of medical management such as treatment of motor function, postural deformity, and epilepsy. But, careful assessment seems necessary to direct management appropriately. It is important that appropriate treatment is found and evaluated, to improve the well-being of these children. Experimental and clinical studies suggest that afferent inputs in somatic nerve fibres have a significant effect on autonomic reflexes, pain and visceral disorders.27 Physiological counterparts can be seen in physical exercise, and the effect can be reproduced artificially via various types of electrical or manual stimulation to certain nerve fibres.28 Some disorders and disabilities may limit or prevent the children from performing certain types of physical exercises. Therefore, different methods, such as acupuncture, which aim to activate sensory afferents may be provide as complement or alternative treatment in these children. Acupuncture has a strong vasodilatory effect 29,30 and affects pain control. 28 It has also been noticed that in many patients acupuncture induces an increased sense of well-being, calmness and improved sleep. Other sensory stimulation methods are transcutaneous electrical nerve stimulation (TENS), vibratory stimulation and massage and all induce excitation of receptors or nerve fibres in the stimulated tissue.28 However, at present there are inconsistencies in the literature with regard to the effects of different sensory stimulation techniques on peripheral blood flow and skin temperature in patients and healthy adults which show the importance of continuing work in this field.31–35 In the future, there is a need to elucidate the role of sympathetic nerve activity in mechanistic studies and also to evaluate interventions with the aim to modulate sympathetic nerve activity.
5.4.
Proxy report
The response rate in this study was 67%, a reasonably good response for postal surveys. There is a risk of respondent bias in that the families who returned the questionnaires might not be representative of the entire group. The number of responses may be influenced by factors such as whether the parents perceive it relevant to provide the information. We do not know whether non-responders were unable to interpret the questionnaire or simply chose not to reply.
1 2 (20 08) 89 – 9 6
95
Compared to an indirect assessment of a person by parents, family members, and caregivers, the assumption is that the child itself is the truest source of information.36 But a low level of agreement between proxies and children is predominately seen when dealing with subjective dimensions such as social and emotional functioning and psychological domains compared with more observable aspects such as physical activity, functioning, and symptoms.36 Further, it was important to obtain parental reports for all children in order to include all children with CP, even those children who could not express themselves, to make comparisons of accompanying impairments and symptoms across the range of abilities. Despite obvious limitations and potential biases of proxy assessments, this report provides at least a partial view of the child’s condition.
6.
Conclusion
Besides the accompanying impairments, a large proportion of the children with CP were reported to have cold extremities, pain, sleeping disorders, constipation, and impaired wellbeing. Most of them had had one or several of these five symptoms for more than 1 year, but their condition was largely untreated. A higher frequency of the symptoms assessed in this study was generally reported in non-walking children than in walkers. Although pain, constipation, and sleeping disorders may have different underlying causes in children with CP, these symptoms may be mediated or accentuated by dysfunction of the autonomic nervous system. To improve the child’s well-being, early recognition of significant accompanying symptoms is important, and adequate treatment is needed.
Acknowledgements This study was funded by the Department of Research and ¨ rnsko¨ldsvik Hospital, O ¨ rnsko¨ldsDevelopment, Sundsvall; O vik; and the Norrbacka-Eugeniastiftelsen in Sweden. We also thank colleagues and parents at the participating habilitation departments for valuable assistance. R E F E R E N C E S
[1] Himmelmann K, Hagberg G, Beckung E, Hagberg B, Uvebrant P. The changing panorama of cerebral palsy in Sweden. IX. Prevalence and origin in the birth-year period 1995–1998. Acta Paediatr 2005;94(3):287–94. [2] Bax M, Goldstein M, Rosenbaum P, Leviton A, Paneth N, Dan B, et al. Proposed definition and classification of cerebral palsy, April 2005. Dev Med Child Neurol 2005;47(8):571–6. [3] Krageloh-Mann I, Hagberg G, Meisner C, Schelp B, Haas G, Eeg-Olofsson KE, et al. Bilateral spastic cerebral palsy—a comparative study between south-west Germany and western Sweden, I: clinical patterns and disabilities. Dev Med Child Neurol 1993;35(12):1037–47. [4] Pharoah PO, Cooke T, Johnson MA, King R, Mutch L. Epidemiology of cerebral palsy in England and Scotland, 1984–9. Arch Dis Child Fetal Neonatal Ed 1998;79(1):F21–5.
ARTICLE IN PRESS 96
E U R O P E A N J O U R N A L O F PA E D I AT R I C N E U R O L O G Y
[5] Beckung E, Hagberg G. Neuroimpairments, activity limitations, and participation restrictions in children with cerebral palsy. Dev Med Child Neurol 2002;44(5):309–16. [6] Himmelmann K, Beckung E, Hagberg G, Uvebrant P. Gross and fine motor function and accompanying impairments in cerebral palsy. Dev Med Child Neurol 2006;48(6):417–23. [7] Svedberg LE, Stener-Victorin E, Nordahl G, Lundeberg T. Skin temperature in the extremities of healthy and neurologically impaired children. Eur J Paediatr Neurol 2005;9(5): 347–54. [8] Holmes Jr TW. Gilfillan RS, Cuthbertson EM. Sympathectomy in the release of vasoconstriction: similarities of response in cerebral palsy and poliomyelitis. Surgery 1967;61(1):129–35. [9] Svedberg LE, Nordahl UE, Lundeberg TC. Effects of acupuncture on skin temperature in children with neurological disorders and cold feet: an exploratory study. Complement Ther Med 2001;9(2):89–97. [10] Wanklyn P, Ilsley DW, Greenstein D, Hampton IF, Roper TA, Kester RC, et al. The cold hemiplegic arm. Stroke 1994;25(9):1765–70. [11] Sullivan M, Karlsson J, Ware Jr. JE. The Swedish SF-36 Health Survey—I. Evaluation of data quality, scaling assumptions, reliability and construct validity across general populations in Sweden. Soc Sci Med 1995;41(10):1349–58. [12] Palisano R, Rosenbaum P, Walter S, Russell D, Wood E, Galuppi B. Development and reliability of a system to classify gross motor function in children with cerebral palsy. Dev Med Child Neurol 1997;39(4):214–23. [13] Morris C, Bartlett D. Gross Motor Function Classification System: impact and utility. Dev Med Child Neurol 2004;46(1):60–5. [14] Morris C, Galuppi BE, Rosenbaum PL. Reliability of family report for the Gross Motor Function Classification System. Dev Med Child Neurol 2004;46(7):455–60. [15] Karlberg J, Luo ZC, Albertsson-Wikland K. Body mass index reference values (mean and SD) for Swedish children. Acta Paediatr 2001;90(12):1427–34. [16] Korpelainen JT, Sotaniemi KA, Myllyla VV. Hyperhidrosis as a reflection of autonomic failure in patients with acute hemispheral brain infarction. An evaporimetric study. Stroke 1992;23(9):1271–5. [17] Korpelainen JT, Sotaniemi KA, Myllyla VV. Asymmetrical skin temperature in ischemic stroke. Stroke 1995;26(9):1543–7. [18] Chelimsky G, Chelimsky T. Familial association of autonomic and gastrointestinal symptoms. Clin Auton Res 2001;11(6):383–6. [19] Hamilton B. An overview of proposed mechanisms underlying thermal dysfunction. Thermology 1985;1:81–7. [20] Ja¨nig W. Functions of the sympathetic innervation of the skin. In: Loewy A, Spyer K, editors. Central regulation of
[21]
[22]
[23]
[24]
[25]
[26]
[27]
[28]
[29]
[30]
[31]
[32]
[33] [34] [35] [36]
12 (2008) 89 – 96
autonomic functions. New York, NY: Oxford University Press; 1990. p. 334–48. Houlihan CM, O’Donnell M, Conaway M, Stevenson RD. Bodily pain and health-related quality of life in children with cerebral palsy. Dev Med Child Neurol 2004;46(5):305–10. Del Giudice E, Staiano A, Capano G, Romano A, Florimonte L, Miele E, et al. Gastrointestinal manifestations in children with cerebral palsy. Brain Dev 1999;21(5):307–11. Staiano A, Del Giudice E. Colonic transit and anorectal manometry in children with severe brain damage. Pediatrics 1994;94(2 Pt 1):169–73. Jan JE, Freeman RD. Melatonin therapy for circadian rhythm sleep disorders in children with multiple disabilities: what have we learned in the last decade? Dev Med Child Neurol 2004;46(11):776–82. Yang TF, Chan RC, Kao CL, Chiu JW, Liu TJ, Kao NT, et al. Power spectrum analysis of heart rate variability for cerebral palsy patients. Am J Phys Med Rehab 2002;81(5):350–4. Bronnikov VA, Kravtsov YI. Non-specific brain systems and the rehabilitation of children with cerebral palsy. Neurosci Behav Physiol 2006;36(9):1003–8. Sato A, Sato Y, Schmidt RF. The impact of somatosensory input on autonomic functions. Rev Physiol Biochem Pharmacol 1997;130:1–328. Andersson S, Lundeberg T. Acupuncture—from empiricism to science: functional background to acupuncture effects in pain and disease. Med Hypotheses 1995;45(3):271–81. Jansen G, Lundeberg T, Kjartansson J, Samuelson UE. Acupuncture and sensory neuropeptides increase cutaneous blood flow in rats. Neurosci Lett 1989;97(3):305–9. Lundeberg T. Peripheral effects of sensory nerve stimulation (acupuncture) in inflammation and ischemia. Scand J Rehab Med Suppl 1993;29:61–86. Balogun JA, Biasci S, Han L. The effects of acupuncture, electroneedling and transcutaneous electrical stimulation therapies on peripheral haemodynamic functioning. Disabil Rehab 1998;20(2):41–8. Calmels P. A scientific perspective on developing acupuncture as a complementary medicine. Disabil Rehab 1999;21(3):129–30 discussion 137–8. Hirokawa S. Acupuncture: neurophysiological perspectives. Disabil Rehab 1999;21(3):131–132, discussion 137–8. Allison GT, Singer KP. Critical perspectives. Disabil Rehab 1999;21(3):133 discussion 137–8. Newham DJ. Methodological perspectives. Disabil Rehab 1999;21(3):134–6 discussion 137–8. White-Koning M, Arnaud C, Bourdet-Loubere S, Bazex H, Colver A, Grandjean H. Subjective quality of life in children with intellectual impairment—how can it be assessed? Dev Med Child Neurol 2005;47(4):281–5.