ArticlePDF Available

Cerebral palsy in Moldova: subtypes, severity and associated impairments

Authors:

Abstract and Figures

Background: Moldova is ranked as one of the countries in Europe with the lowest income per capita and with a relatively high infant and maternal mortality rate. Information on neurodisabilities in general is limited, and regarding cerebral palsy (CP) in particular, it is completely lacking. The aim of this study was therefore to make a crude estimate of the prevalence of CP and to describe subtypes and the severity of motor impairments and associated problems in this country. Methods: Children with CP born 2009-2010, attending the National Hospital Institute of Mother and Child, the reference hospital for ~ 75% of children in Moldova with neurological disabilities, were identified from medical records. Results: Among 207 children with CP (estimated prevalence 3.4 per 1000 live births), 185 (mean age 7.3 years; 36% girls) had detailed information. Thirty seven (20%) children had spastic unilateral, 113 (61%) spastic bilateral, 22 (12%) dyskinetic and 9 (5%) children had ataxic CP. The subtype was unclassified in four children. Among all children, 93 (51%) had epilepsy, 109 (59%) intellectual disability, 42 (23%) severe vision and 10 (5%) hearing impairments while 84 (45%) children had severe speech impairments. Fifty-two (28%) children were born prematurely, and 46 (25%) had Apgar scores below 7 at five minutes. Conclusion: Compared to other European studies, the distribution of CP subtypes was different in Moldova. Moreover, the estimated prevalence, the proportions with severe motor and associated impairments and of children born at term were higher in Moldova while the proportion with low Apgar did not differ. The findings may suggest different etiological pathways causing CP in Moldova than in other European countries. A national register is warranted for quality assurance and improvement.
Content may be subject to copyright.
R E S E A R C H A R T I C L E Open Access
Cerebral palsy in Moldova: subtypes,
severity and associated impairments
Ecaterina Gincota Bufteac
1,2*
, Guro L. Andersen
3,4
, Vik Torstein
4
and Reidun Jahnsen
5
Abstract
Background: Moldova is ranked as one of the countries in Europe with the lowest income per capita and with a
relatively high infant and maternal mortality rate. Information on neurodisabilities in general is limited, and
regarding cerebral palsy (CP) in particular, it is completely lacking. The aim of this study was therefore to make a
crude estimate of the prevalence of CP and to describe subtypes and the severity of motor impairments and
associated problems in this country.
Methods: Children with CP born 20092010, attending the National Hospital Institute of Mother and Child, the
reference hospital for ~ 75% of children in Moldova with neurological disabilities, were identified from medical
records.
Results: Among 207 children with CP (estimated prevalence 3.4 per 1000 live births), 185 (mean age 7.3 years; 36%
girls) had detailed information. Thirty seven (20%) children had spastic unilateral, 113 (61%) spastic bilateral, 22
(12%) dyskinetic and 9 (5%) children had ataxic CP. The subtype was unclassified in four children. Among all
children, 93 (51%) had epilepsy, 109 (59%) intellectual disability, 42 (23%) severe vision and 10 (5%) hearing
impairments while 84 (45%) children had severe speech impairments. Fifty-two (28%) children were born
prematurely, and 46 (25%) had Apgar scores below 7 at five minutes.
Conclusion: Compared to other European studies, the distribution of CP subtypes was different in Moldova.
Moreover, the estimated prevalence, the proportions with severe motor and associated impairments and of
children born at term were higher in Moldova while the proportion with low Apgar did not differ. The findings may
suggest different etiological pathways causing CP in Moldova than in other European countries. A national register
is warranted for quality assurance and improvement.
Keywords: Cerebral palsy, Moldova, Epidemiology, Subtype, Severity
Background
Cerebral palsy (CP) describes a group of permanent disor-
ders of the development of movement and posture, causing
activity limitations that are attributed to non-progressive
disturbances occurring in the developing foetal or infant
brain. The motor impairments are often accompanied by
disturbances of sensation, perception, cognition, communi-
cation, and behaviour, by epilepsy and by secondary muscu-
loskeletal problems [1].
Considering the significant differences in perinatal and
infant mortality between high- and low-income countries,
it may seem paradoxical that the prevalence of CP has
been reported to be similar, at around 23 per 1000 live
births both in developing countries [2,3] and in developed
countries [4,5]. However, the clinical manifestations of
CP differ significantly between low- and high income
countries [3,6,7], and some studies have reported that
perinatal asphyxia and hyperbilirubinaemia are more com-
mon causes in developing countries [2,3,7] . In studies
describing the panorama of CP, ascertainment of cases is a
challenge even in developed countries [5]. In this regard,
it is noteworthy that other studies in developing countries
have reported CP prevalence between 3.4 and 4.5 per
1000 live births [3,7].
* Correspondence: kbufteac@gmail.com;s307865@hioa.no;voinicel@usmf.md
1
Department of Health Sciences, Oslo Metropolitan University, PO box 4 St.
Olavs plass, No-0130 Oslo, Norway
2
Early Intervention Center Voinicel, Drumul Taberei str., nr.2A, Chisinau,
Moldova
Full list of author information is available at the end of the article
© The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver
(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332
https://doi.org/10.1186/s12887-018-1305-6
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
Moldova is ranked as the country in Europe with the
lowest gross domestic product (GDP) i per capita, esti-
mated to around USD 5700 in 2017, compared with an
average of USD 40000 for the European Union. Thus, fi-
nancial resources for advanced medical care are limited,
even if the proportion of the GDP spent for health ex-
penditures (10%) is comparable to the proportion spent
by the Nordic countries. These limited financial re-
sources are a likely explanation of the high maternal
mortality rate of 23 deaths per 100,000 live births; five to
six times higher than in the Nordic countries, and of the
infant mortality rate of 12 deaths per 1000 live births,
being more than twice as high as in the Nordic countries
(9). The prevalence of CP and distribution of CP sub-
types, motor impairments and associated problems is
unknown, and access to orthopaedic surgery as well as
to modern treatment, such as Botulinum toxin injections
are very limited (personal observation) [8,9].
Thus, the aim of this study is to describe the pano-
rama of CP in Moldova with emphasis on CP subtypes,
the severity of the motor impairments and the occur-
rence of associated impairments, as well as how these
clinical characteristics relate to perinatal risk factors.
Methods
Design and study population
Eligible for inclusion in this cross-sectional study were
children with CP treated at the National Hospital Institute
of Mother and Child, in Chisinau, Moldova, born between
January 1st 2009 and December 31st 2010. This hospital is
the reference hospital for children with disabilities living
outside the Capital area, covering approximately 2.7
million (75%) of Moldovas 3.5 mill inhabitants. Acquired
CP lesions after the neonatal period were excluded. Chil-
dren with neurological impairments are referred to this
hospital every six months for examination and treatment.
Children diagnosed with CP were identified by the first
author via medical records from the Departments of
child neurology (318 years) and of toddler neurology
(3 months to 3 years). The data were collected between
June and September 2016, when the children were 7
8 years old. Detailed information regarding neurological
status, motor function and associated impairments was
identified by scrutinizing the medical records of each
child from several departments. Antenatal and perinatal
information was collected from the medical records at
the Maternity ward, and at the Departments of neonatal
care, premature birth and from the Neonatal Intensive
Care Unit (NICU).
Variables
CP was diagnosed and classified consistent with the rec-
ommendations proposed by the Surveillance of Cerebral
Palsy in Europe (SCPE) [10] into spastic, dyskinetic,
ataxic and not classified subtypes. The spastic subtype
was sub-classified into unilateral and bilateral subtypes,
spastic unilateral CP into right and left hemiplegia, and
spastic bilateral CP into quadriplegia and diplegia in ac-
cordance with the International Classification of Disease
10 (ICD 10) [11].
Gross motor function was classified according to the
Gross Motor Function Classification System (GMFCS)
[12] using the available information on walking and sit-
ting abilities. Fine motor function was classified accord-
ing to the Bimanual Fine Motor Function (BFMF) scale
using the available information on hand function [13].
As the data were collected from medical records of vari-
able quality, the childrens gross motor function was cat-
egorized into those who were able to walk without
assistive devices (corresponding to GMFCS level I and
II), those who were able to walk with assistive devices
corresponding to (GMFCS level III) and those who were
unable to walk even with such devices (corresponding to
GMFCS levels IV-V).
Fine motor function is described by speech therapists
in the free text of medical records. Based upon these de-
scriptions, fine motor function was classified according
to BFMF, as proposed by Andersen et al. [4]. As for
GMFCS, the BFMF classification was reduced to three
categories; i.e. BFMF level I-II, BFMF level III and BFMF
levels IV-V [4].
Associated impairments
Speech therapists tested cognitive abilities in the hospital
and described speech and feeding abilities in free text in
the medical records.
Cognitive function was assessed using the Development
Assessment of Young Children Evaluation tool (DAYC)
and classified into intellectual disability (intelligence quo-
tient (IQ) score < 70) and normal intellectual ability (IQ
score 70) [14].
Speech was classified on a five-level scale from zero to
four, where zero indicated normal speech, level I indicated
indistinct speech, level II obviously indistinct speech, level
III severely indistinct speech (difficult to understand) and
level IV indicated children with no speech.
Feeding ability was classified into five levels, 0 indicat-
ing that the child did not have feeding problems, level I
indicating that the child was in need of some assistance,
level II that the child was entirely dependent on assist-
ance, level III that the child was partly tube fed and level
IV that the child was mainly tube fed. No information
on gastrostomy was recorded. The five levels were then
dichotomised into two categories: independent feeding
and dependent on caregiver assistance.
Vision was described as 0 for normal, 1 for impaired
and 2 for severely impaired [10], and later dichotomised
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 2 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
into 0 (01 normal or impaired) and 1 (2 severely
impaired).
Hearing was described as 0 for normal, 1 for impaired
and 2 for severely impaired, and later dichotomized into 0
(01 normal or impaired) and 1 (2 for severely impaired).
Epilepsy was defined as two unprovoked seizures, ex-
cluding neonatal and febrile seizures. Epilepsy was con-
sidered active if the child used antiepileptic drugs at the
time of the registration.
Perinatal data
Available perinatal data included gestational age (GA),
birth weight (BW) and Apgar scores at one and five mi-
nutes. Gestational age at birth is usually estimated from
ultrasonography at GA week 1822. In this study, GA
was categorized into extremely low (ELGA < week 28,
very low (VLGA: GA week 28 (28.0) to < 32 (31 weeks;
six days), moderately low (MLGA: GA week 32 (32.0) to
37 (36 weeks, six days) and term-born (GA week 37.0 or
later). Birth weight was recorded to the nearest gram (g)
and was categorized as extremely low (ELBW < 1000 g);
very low (VLBW: 10001499 g); moderately low
(MLBW: 15002499 g) and normal (BW > 2500 g).
Apgar scores at one and five minutes were categorized
into three categories: 03, 46 and 710.
Statistical methods
The Statistical Package for Social Sciences (IBM SPSS
V.19) was used for data analysis, and a significance level of
0.05 was chosen. Descriptive statistics with frequency ta-
bles were performed to show proportions of subtypes and
CP severity levels, and the proportion of children with as-
sociated impairments. Differences in proportions were
evaluated using Chi-square statistics, and p-values < 0.05
were considered statistically significant.
Ethics
The study was approved by the National Committee for
Ethical Expertise in Clinical Trials (Nr. 266) and by the
Centre of Early Intervention VoinicelEthical Commit-
tee (nr. 01/17). A statement of Agreement of Collabor-
ation between the State Medical and Pharmacy
University and the Institute of Mother and Child was
signed, and finally, permission to conduct the study was
obtained from the Ministry of Health (12th of July, nr.
08/88), from the Municipality Council of Social Protec-
tion, (13.07.2016, nr. 070117/957) and from the Ministry
of Labour, Social Protection and Family of the Republic
of Moldova.
Permission to access the childrens data was obtained
from the Republican Ethics Committee, The State Med-
ical and Pharmacy Ethics Committee, Ministry of Health
and Ministry of Labour and Social Protection and the
Directors of the Main Hospital for Children in RM- In-
stitute of Mother and Child.
Parents or primary caregivers of patients treated at this
centre sign an informed consent form stating that med-
ical data collected as part of the admission may be used
for research purposes. The use of this regularly collected
medical information in the present study was approved
as described above and did not require a new individual
consent form to be completed.
Results
In all, 207 children with CP were identified. The total
number of live births during 2009 and 2010 in Moldova
was 81,277, and a crude estimate of CP prevalence
would be 3.4 per 1000 live births if the children in this
study were recruited from 75% of the birth population.
Of the 207 children, 22 had incomplete data. Thus, the
study population comprised 185 children with a mean
age of 7.3 years (range: 6.67.6 years) and 67 (36%) of
these were girls. Thirty-seven children (20%) had spastic
unilateral, 113 (61%) had spastic bilateral, 22 (12%) had
dyskinetic and nine (5%) had ataxic CP. In four children
(2%), the subtype could not be classified. Mean age at
diagnosis was 23 months (range: 12-91 months). Among
children with unilateral spastic CP, the extremities of the
right side were affected in 21 (57%) and on the left side
in 16 (43%) (p= 0.502). Among those with spastic bilat-
eral CP, 19 (17%) had diplegic, while 94 (83%) had
quadriplegic CP (p= 0.001).
Table 1shows that 60 (31%) children were able to walk
independently (GMFCS level I-II), 53 children (33%)
were able to walk with assistive devices (GMFCS level
III) and the remaining 72 (36%) children were unable to
walk (GMFCS level IV-V) (p= 0.001). Table 1also shows
that 73 (40%) of the children had hand function corre-
sponding to BFMF level I or II, 47 (24%) had BFMF level
III and 65 (36%) had hand function corresponding to
level IV or V (p= 0.001). Children with unilateral CP
had better gross and fine motor function compared to
children with bilateral and dyskinetic CP (p= 0.001)
(Table 1).
Associated impairments
Table 1shows that 109 (59%) children had an intellec-
tual disability. In total, 42 (23%) had severe visual and
ten (5%) severe hearing impairment, 93 (50%) had active
epilepsy, 84 (45%) had severely impaired speech or no
speech and 80 (43%) children were unable to eat inde-
pendently (p= 0.001). Ten (5%) of the 185 children with
CP were severely affected by all of these impairments.
Table 1also shows that associated impairments were
most prevalent among children with dyskinetic and
spastic bilateral CP subtypes (p= 0.001), while they were
least prevalent among children with unilateral CP (p=
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 3 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
0.041). Among children with unilateral CP, there were
no significant differences in the prevalence of associated
impairments between children with right- or left-sided
affection (p> 0.056). All ten children who were severely
affected by all associated impairments had the spastic bi-
lateral subtype. None of the 84 children with severe
speech impairment communicated with the help of aug-
mentative or alternative communication aids (i.e. pic-
tures or pictograms). The proportion of children without
any associated impairments in this study was 11%.
Perinatal risk factors
In total, 133 (72%) children with CP were born at
term, whereas 10 (5%) were born ELGA. Among chil-
dren born at term and preterm, the most common
subtype was spastic bilateral CP, accounting for 54%
and 79%, respectively (Table 2). Only one of 23
children born before GA week 32 was able to walk
without assistive devices (GMFCS level I-II), com-
pared to 11 (38%) children born moderately preterm,
and 48 (36%) children born at term (p=0.023). Five
(22%) of the 23 children born before week 32 had
good hand function (BFMF I-II); however, this pro-
portion was lower when compared to those born at
term, 54 (41%) of 133 children born 37 weeks had
good hand function (p< 0.001) (Table 3).
Table 4shows that epilepsy was present in a higher
proportion of children born at term compared to those
born preterm, while intellectual disability, severe vision
and hearing impairments were more prevalent in chil-
dren born before GA 28 weeks compared to those born
at term (p= 0.024).
A total of 130 (70%) children had normal BW, and six
(3%) had BW < 1000 g. Among children with normal
Table 1 Subtypes, severity and associated impairments in a cohort of 185 children with CP
CP subtypes
Unilateral N (%) Bilateral N (%) Dyskinetic N (%) Ataxic N (%) Not classified N (%) Total N (%) P-value of totals
GMFCS levels
Level I-II 33 (89) 21 (19) 0 5 (56) 1 (25) 60 (33)
Level III 4 (11) 39 (35) 4 (18) 4 (44) 2 (50) 53 (29)
Level IV-V 0 53 (28) 18 (82) 0 1 (25) 72 (38) < 0.001
BFMF levels
Level I-II 33 (91) 35 (31) 0 4 (44) 1 (25) 73 (40)
Level III 4 (10) 32 (28) 4 (18) 5 (56) 2 (50) 47 (24)
Level IV-V 0 46 (41) 18 (82) 0 1 (25) 65 (36) < 0.001
Intellectual disability
No 31 (84) 39 (35) 2 (9) 3 (34) 1 (25) 76 (41)
Yes 6 (16) 74 (66) 20 (91) 6 (67) 3 (75) 109 (59) 0.001
Active epilepsy
No 29 (78) 48 (43) 8 (36) 3 (33) 4 (100) 92 (49)
Yes 8 (22) 65 (58) 14 (64) 6 (67) 0 93 (51) < 0.001
Vision
Normal/impaired 34 (92) 80 (71) 19 (86) 7 (78) 3 (75) 143 (77)
Severely impaired 3 (8) 33 (29) 3 (14) 2 (22) 1 (25) 42 (23) 0.054
Hearing
Normal/impaired 37 (100) 104 (92) 21 (96) 9 (100) 4 (100) 175 (95)
Severely impaired 0 9 (8) 1 (5) 0 0 10 (5) 0.143
Speech impairments
Normal 11 (30) 8 (7) 1 (5) 0 0 20 (11)
Impaired, understandable 25 (68) 43 (38) 7 (32) 5 (56) 1 (25) 81 (44)
Severely impaired/no speech 1 (3) 62 (55) 14 (64) 4 (44) 3 (75) 84 (45) < 0.001
Feeding
Independent 34 (92) 56 (49) 8 (36) 5 (56) 2 (50) 105 (57)
Dependent 3 (8) 57 (51) 14 (64) 4 (44) 2 (50) 80 (43) < 0.001
Total 37 (100) 113 (100) 22 (100) 9 (100) 4 (100) 185 (100)
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 4 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
BW, the most common CP subtype was spastic bilateral,
72 (64%) (p< 0.001) (Table 2).
Finally, 79 (43%) children had a low Apgar score (03
and 46) at one and 46 (25%) at five minutes (Table 3).
Low Apgar scores were associated with more severe im-
pairments of gross and fine motor function (p= 0.029)
and with spastic bilateral and dyskinetic CP (p= 0.057)
(Table 3).
Discussion
In this first study of the panorama of CP in Moldova we
found a crude prevalence estimate of 3.4 per 1000 live
births and that spastic bilateral CP was the most com-
mon subtype, accounting for nearly two-thirds of all
cases in this population of children. Only every fifth
child had the spastic unilateral CP subtype, and only one
third could walk without an assistive device. Most of the
children had significantly impaired fine motor function,
and associated problems were common. Almost half of
the children had barely understandable or no speech
and a concern is that none of them used assisted or aug-
mented communication. Only 28% of the children were
born prematurely, and around 10% of the children were
born at GA below 32 weeks, or with BW below 1500 g.
Every fourth child had Apgar scores below 7 at five
minutes.
Validity
Strengths and limitations
The strength of the study is the hospital-based cohort
from the main paediatric hospital of Moldova Institute
of Mother and Child. As this hospital cares for all chil-
dren living outside the Municipality of Chisinau, the co-
hort may be considered population-based and
representative of 75% of the population of Moldova. The
regular assessment of the children provided high-quality
clinical information that could be systematised. The
mean age of the children when the study was performed
was 7.3 years old, which represents a strength regarding
the validity of the CP diagnosis [9,15]. Access to a var-
iety of medical records also ensured ascertainment of
the cases. Nonetheless, we cannot exclude that some
children with milder forms of CP (for example, some
children with unilateral spastic CP with GMFCS and
BFMF level I), were not referred to this central hospital.
Such selection bias could, therefore, have contributed
somewhat to the low proportion of children with the
unilateral CP subtype and the high proportion of chil-
dren with severe motor impairments and associated
problems. In contrast, the crude estimate of prevalence
was high, and even in the most unlikely event that the
cases included in this study would represent all children
with CP in Moldova, the prevalence would be 2.5 per
1000 live births. The latter figure would still be high
Table 2 Children with different CP subtypes by gestational age (GA), birth weight (BW) and Apgar score
CP subtypes
Unilateral N (%) Bilateral N (%) Dyskinetic N (%) Ataxic N (%) Not classified N (%) Total N (%) P-value of totals
GA
GA < 28 weeks 1 (3) 7 (6) 1 (5) 0 1 (25) 10 (5)
GA 2831 weeks 0 11 (10) 0 1 (11) 0 12 (7)
GA 3236 weeks 4 (11) 23 (20) 2 (9) 1 (11) 0 30 (16)
GA 37 weeks 32 (86) 72 (64) 19 (86) 7 (78) 3 (75) 133 (72) 0.002
BW
BW < 1000 g 0 6 (5) 0 0 0 6 (3)
BW 1000-1499 g 1 (3) 8 (7) 1 (5) 0 1 (25) 11 (6)
BW 1500-2499 g 5 (14) 27 (24) 4 (18) 1 (11) 1 (25) 38 (21)
BW 2500 g 31 (83) 72 (64) 17 (77) 8 (89) 2 (50) 130 (70) 0.054
Apgar (1 min)
Apgar 03 1 (3) 19 (17) 1 (5) 1 (11) 1 (25) 23 (12)
Apgar 46 8 (22) 40 (35) 5 (23) 2 (22) 1 (25) 56 (30)
Apgar 710 28 (75) 54 (48) 16 (72) 6 (67) 2 (50) 106 (58) 0.057
Apgar (5 min)
Apgar 03 1 (3) 7 (6) 2 (9) 1 (11) 0 11 (6)
Apgar 46 1 (3) 30 (27) 2 (9) 0 2 (50) 35 (19)
Apgar 710 35 (94) 76 (67) 18 (82) 8 (89) 2 (50) 139 (75) 0.044
Total 37 (100) 113 (100) 22 (100) 9 (100) 4 (100) 185 (100)
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 5 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
compared with other European countries [5,16,19].Thus,
we consider it unlikely that the main findings and conclu-
sions in this study can be completely explained by selec-
tion bias.
Another limitation is that the classification of gross
and fine motor function (GMFCS and BFMF) was made
in retrospect, based upon text descriptions in the med-
ical records. However, the main criterion in the classifi-
cation of GMFCS is the ability to walk with or without
assistive devices or not at all. Therefore, we believe that
misclassification of GMFCS is unlikely to explain the re-
sults regarding gross motor function. The classification
of fine motor function (BFMF) according to Andersen et
al. [4] is more complicated. Interestingly, the proportion
of children with severely impaired fine motor function
in this study is about the same as Andersen et al. [4].
However, it should be noted that fine motor function
was found to be more impaired in Norway compared to
other populations at that time. Thus, the findings for
fine motor function in this study should be interpreted
with caution. Assessments of cognitive abilities, speech
and feeding were performed by experienced speech ther-
apists, mostly using validated instruments, which is a
strength of this study.
Comparison with the literature
In the present study the distribution of spastic and
non-spastic CP subtypes is comparable to other studies
in Europe [4,16].
However, the proportion of children with unilateral
spastic CP is lower, while spastic bilateral (particularly
quadriplegic CP) and dyskinetic forms were higher in
our study compared to several studies in Europe and
Australia [4,1618].
Among term born children, the proportion with dyski-
netic CP in our study (12%) was lower compared with
Himmelmann et al. (23%) [19], but higher than in two
other studies [4,19]. The percentage of children with
the ataxic subtype is similar to other studies [4].
The distribution of CP subtypes among children born
preterm in this study was comparable to the distribution
reported by Himpens et al. [16] in their metanalysis of
studies including children born later than 1980.
An important finding is that the proportion of chil-
dren who were able to walk independently (33%) was
lower in the present study than in a Norwegian study
(55%), Andersen et al. [4], and in the majority of the
studies included in the meta-analysis by Himpens et
al. [16].
Table 3 Severity of gross and fine motor impairments in children with CP by gestational age (GA), birth weight (BW) and Apgar
score
Estimated GMFCS Estimated BFMF
I-II N (%) III N (%) IV-V N (%) Total N (%) I-II N (%) III N (%) IV-V N (%) Total N (%)
GA
GA < 28 weeks 1 (2) 5 (9) 5 (7) 11 (6) 4 (6) 3 (6) 4 (6) 11 (6)
GA 2831 weeks 0 7 (13) 5 (7) 12 (7) 1 (1) 7 (15) 4 (6) 12 (7)
GA 3236 weeks 11 (18) 11 (21) 7 (10) 29 (15) 14 (19) 9 (19) 6 (10) 29 (15)
GA > 37 weeks 48 (80) 30 (57) 55 (76) 133 (72) 54 (74) 28 (60) 51 (78) 133 (72)
BW
BW < 1000 g 1 (2) 3 (6) 2 (3) 6 (3) 3 (4) 1 (1) 2 (3) 6 (3)
BW 1000-1499 g 2 (3) 4 (7) 5 (7) 11 (6) 3 (4) 4 (9) 4 (6) 11 (6)
BW 1500-2499 g 10 (17) 12 (23) 16 (22) 38 (21) 12 (17) 13 (28) 13 (20) 38 (21)
BW > 2500 g 47 (78) 34 (64) 49 (68) 130 (70) 55 (75) 29 (62) 46 (71) 130 (70)
Apgar (1 min)
Apgar 03 1 (1) 7 (13) 15 (21) 23 (13) 3 (5) 7 (15) 13 (20) 23 (13)
Apgar 46 16 (27) 16 (30) 24 (33) 56 (30) 20 (27) 16 (34) 20 (31) 56 (30)
Apgar 710 43 (72) 30 (57) 33 (46) 106 (57) 50 (68) 24 (51) 32 (49) 106 (57)
Apgar (5 min)
Apgar 03 1 (2) 3 (5) 7 (10) 11 (6) 1 (2) 4 (9) 6 (9) 11 (6)
Apgar 46 6 (10) 11 (21) 18 (25) 35 (19) 9 (12) 10 (21) 16 (25) 35 (19)
Apgar 710 53 (88) 39 (74) 47 (65) 139 (75) 63 (86) 33 (70) 43 (66) 139 (75)
Total 60 (100) 53 (100) 72 (100) 185 (100) 73 (100) 47 (100) 65 (100) 185 (100)
GMFCS Gross Motor Function Classification System estimated from descriptions of walking and sitting abilities
BFMF Bimanual Fine Motor Function estimated from descriptions of hand function in each hand separately
P-value GMFCS/GA,BW,Apgar 1,5 min < 0.002
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 6 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
The proportion of children without any associated im-
pairments was 11% in this study compared to 28% in
Norway [4] and 48% in Sweden [19]. However, the latter
study did not include speech impairments [19]. When
we excluded speech impairments, we found that 35% of
the Moldovan children with CP had no associated im-
pairments (Table 4), which is comparable to results from
Norway (34%) [4], but lower than in Sweden and
Deutschland [19,20].
The proportion of children with severely impaired
or no speech in our study was higher than in a Nor-
wegian study [4]. This finding is reasonable, consider-
ing the low proportion of children with unilateral
spastic CP, and the high proportion of children with
severe gross motor impairments (GMFCS level IV-V)
in our study. However, it is concerning that none of
the children with severely impaired speech had any
form of assisted or alternative communication (ASC).
This finding may be partially consistent with one
study in Norway reporting that approximately 50% of
children in need of such communication did not use
ASC [21].
The proportions of children born prematurely, and
born before week 28 were lower in Moldova than in two
Nordic studies [4,19]. This may be partly explained by
lower proportions in the background population (0.27%
in Moldova versus 0.4% in Norway) [22], but more likely
by differences in perinatal and neonatal mortality. The
majority of children with CP were born at term, consist-
ent with most other studies.
Despite the higher proportions of children born at
term and with quadriplegic and dyskinetic CP in this
study, the proportion with low Apgar scores was similar
to that reported by Andersen et al. [4] and Himmelmann
et al. [19].
The most severe gross and fine motor impairments were
associated with low Apgar scores. Moreover, children born
with normal BW had more severe motor impairments in
comparison with children with low BW (2500 g). These
findings are consistent with another study conducted in
Moldova between 2008 and 2014 [23].
Interpretation and implications
The lower proportion of children with unilateral CP in
this study, as well as the lower proportion of children able
to walk independently (GMFCS level I-II) could partly be
explained by selection bias. It is possible that families liv-
ing in the most rural areas of Moldova who have a child
with mild unilateral CP are less likely to apply for or be re-
ferred to follow-up at the Institute of Mother and Child.
However, if there are cases missing, the prevalence of CP
must be significantly higher than our estimate. Thus, one
implication of this study is that there is a need to establish
national high-quality health registers in general, and CP
Table 4 Associated impairments by gestational age (GA) in children with CP
GA < 28 N (%) GA 2831 N (%) GA 3236 N (%) GA > 37 N (%) Total N (%) P-value of totals
Intellectual disability
No (> 70) 3 (30) 4 (33) 14 (47) 55 (41) 76 (41)
Yes (< 70) 7 (70) 8 (67) 16 (53) 78 (59) 109 (59) 0.553
Active epilepsy
No 8 (80) 7 (58) 19 (63) 58 (44) 92 (49)
Yes 2 (20) 5 (42) 11 (37) 75 (56) 93 (51) 0.008
Vision
Normal/impaired 6 (60) 9 (75) 24 (80) 104 (78) 143 (77)
Severely impaired 4 (40) 3 (25) 6 (20) 29 (22) 42 (23) 0.303
Hearing
Normal/impaired 9 (90) 12 (100) 30 (100) 124 (93) 175 (95)
Severely impaired 1 (10) 0 0 9 (7) 10 (5) 0.552
Speech Impairments
Normal 1 (10) 0 4 (13) 15 (11) 20 (11)
Impaired, understandable 4 (40) 7 (58) 15 (50) 55 (41) 81 (44)
Severely impaired/No speech 5 (50) 5 (42) 11 (37) 63 (48) 84 (45) 0.994
Feeding
Independent 7 (70) 6 (50) 20 (67) 72 (54) 105 (57)
Dependent 3 (30) 6 (50) 10 (33) 61 (46) 80 (43) 0.347
Total 10 (100) 12 (100) 30 (100) 133 (100) 185 (100)
GA Gestational Age (weeks) in categories (< 28 weeks of gestation, 2831 weeks, 3236 weeks and > 37 weeks of gestation)
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 7 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
registers in particular, in Moldova. This is of the utmost
importance to provide health authorities with correct in-
formation regarding the prevalence and need for interven-
tions but also to monitor ante- and perinatal health.
Notably, a very recent Norwegian study documented the
need for access to several health registers to ensure
complete identification of cases [5].
However, although selection bias may partly explain
the low proportion of children with unilateral CP, we
consider it less likely to be the main explanation of the
difference in CP subtypes between Moldova and other
European studies. First, the crude estimate of prevalence
is higher than in other studies, which makes it unlikely
that there has been a significant underreporting of cases.
Secondly, within the group of children with bilateral CP,
the proportion with quadriplegic CP was very high, and
finally, within the group with bilateral CP, the proportion
able to walk independently was lower in our than in
other populations, Thus, another interpretation may be
related to obstetric and perinatal care. The increased
maternal and infant mortality rates in Moldova com-
pared to Norway, Sweden and Iceland, are most likely
also reflected in higher perinatal and neonatal mortality
rates. The high proportion of children with more severe
manifestations of CP in Moldova may, therefore, also be
explained by less advanced obstetric and neonatal care
in Moldova than in Nordic countries. Both the spastic
quadriplegic and dyskinetic CP subtypes are common in
deliveries complicated by hypoxia-ischemia [24]. How-
ever, low Apgar scores were not more common in this
study than in other studies. Therefore the higher propor-
tion of children with quadriplegic and dyskinetic CP
may have other causes, such as increased prevalence of
pre-pregnancy or pregnancy related maternal disorders,
congenital anomalies [25,26], perinatal infections or pla-
cental disorders. The low proportion of children born
before week 28 could, in part, be explained by different
attitudes and limited access to advanced neonatal inten-
sive care in Moldova. Thus, the other implication of this
study is that there is a need to explore whether obstetric
and neonatal care in Moldova may need to be further
improved to achieve results comparable to other Euro-
pean countries.
Finally, this study has revealed that standard classifica-
tion systems like GMFCS and BFMF, as well as motor
function assessment tools, such as Gross Motor Func-
tion Measurement (version 66 or 88) [12], are not clinic-
ally used on a regular basis. Moreover, assisted or
augmented communication was apparently not used by
children with speech problem. One implication of these
results will, therefore, be to establish a collaborative net-
work with specialists from the rehabilitation centres
throughout the country in order to improve the quality
of care for children with CP in Moldova.
Conclusion
Compared to other populations, children with CP in
Moldova more frequently present with bilateral and dys-
kinetic subtypes. In addition, severe gross motor impair-
ments and associated problems are also more common
in Moldova. Our findings suggest a higher prevalence of
CP, and the causal pathways may be different in
Moldova relative to other European countries. Children
with milder CP symptoms may be less likely to receive
specialist care. These findings suggest a need for a na-
tional health quality register [27], and provide a basis to
improve the quality of perinatal medicine and care for
children with CP.
Abbreviations
BFMF: Bimanual fine motor function; BW: Birth weight; CP: Cerebral palsy;
GA: Gestational age; GDP: Gross domestic product; GMFCS: Gross motor
function classification system; IQ: Intellectual quotient; SCPE: Surveillance of
Cerebral Palsy in Europe; USD: United States dollars
Acknowledgements
We would like to express our thanks to the Ministry of Health and
administrative staff of the Institute of Mother and ChildChisinau, Moldova,
for providing access to the medical records saved in the hospital archives.
Funding
This study was supported by a student grant from the Ministry of Foreign
Affairs of Norway and AHEAD-Moldova, Norway, within the PhD program at
Oslo Metropolitan University, Faculty of Health Sciences, Oslo, Norway.
Availability of data and materials
The datasets generated and analysed during the current study are not
publicly available due to the restrictions to share the data to the third
parties, based on the Management Committee of the Institute of Mother
and Child decision -the hospital which provided the data (written permit
from the Director of the hospital, where its specified that its forbidden to
share the data to the third parties), but are available from the corresponding
author on reasonable request.
Authorscontributions
EGB designed the study, extracted and analysed the data, performed the
statistical analyses, participated in drafting and improving the manuscript. RJ,
GA and TV contributed to the design and conceptualization of the study,
provided critical input and oversight of field work, and reviewed the final
manuscript as submitted. All authors approved the final manuscript as
submitted and agree to be accountable for all aspects of the work.
Authorsinformation
Ecaterina Gincota Bufteac is a medical doctor at Voinicel Early Intervention
Centre in Chisinau, Moldova and PhD candidate at Oslo Metropolitan
University, Norway,
Guro L. Andersen is a pediatrician PhD, leader of the Cerebral Palsy Registry
of Norway (CPRN) at Vestfold Hospital trust and associate professor at
Department of Clinical and Molecular Medicine, Norwegian University of
Science and Technology in Trondheim, Norway.
Vik Torstein is professor of pediatrics at Department of Clinical and Molecular
Medicine, Norwegian University of Science and Technology in Trondheim,
Norway.
Reidun Jahnsen is a physical therapist PhD and senior researcher at Oslo
University Hospital. She is leader of the national CP surveillance program
(CPOP) in Norway.
Ethics approval and consent to participate
The study was approved by the National Committee for Ethical Expertise in
Clinical Trials (Nr. 266) and by the Centre of Early Intervention Voinicel
Ethical Committee (nr. 01/17). A statement of Agreement of Collaboration
between the State Medical and Pharmacy University and the Institute of
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 8 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
Mother and Child was signed, and finally, permission to conduct the study
was obtained from the Ministry of Health (12th of July, nr. 08/88), from the
Municipality Council of Social Protection, (13.07.2016, nr. 070117/957) and
from the Ministry of Labour, Social Protection and Family of the Republic of
Moldova. Permission to access the childrens data was obtained from the
Republican Ethics Committee, The State Medical and Pharmacy Ethics
Committee, Ministry of Health and Ministry of Labour and Social Protection
and the Directors of the Main Hospital for Children in RM- Institute of
Mother and Child.
Consent for publication
not applicable.
Competing interests
The authors declare that they have no competing interests.
PublishersNote
Springer Nature remains neutral with regard to jurisdictional claims in
published maps and institutional affiliations.
Author details
1
Department of Health Sciences, Oslo Metropolitan University, PO box 4 St.
Olavs plass, No-0130 Oslo, Norway.
2
Early Intervention Center Voinicel,
Drumul Taberei str., nr.2A, Chisinau, Moldova.
3
The Cerebral Palsy Register of
Norway (CPRN), Department of Paediatrics, Vestfold Hospital Trust, Tonsberg,
Norway.
4
Department of Clinical and Molecular Medicine, Norwegian
University of Science and Technology, PO Box 8905, 7491 Trondheim,
Norway.
5
The Cerebral Palsy Follow-up Program (CPOP), Department of
Clinical Neurosciences for Children, Oslo University Hospital, Box 4950,
Nydalen, 0424 Oslo, Norway.
Received: 16 March 2018 Accepted: 8 October 2018
References
1. Rosenbaum, P., Paneth, N., Leviton, A., Goldstein, M., Bax, M., Damiano, D., ...
Jacobsson. (2007). A report: the definition and classification of cerebral palsy
April 2006. Dev Med Child Neurol, 49, 814. doi:https://doi.org/10.1111/j.
1469-8749.2007.tb12610.x.
2. El-Tallawy HN, Farghaly WM, Shehata GA, Rageh TA, Metwally NA, Badry R,
Kandil MR. Cerebral palsy in Al-Quseir City, Egypt: Prevalence, subtypes, and
risk factors. Neuropsychiatr Dis Treat. 2014;Vol. 10:126772.
3. Stanley FJ, Blair E, Alberman E. Cerebral palsies: epidemiology and causal
pathways. London: Cambridge University Press; 2000.
4. Andersen GL, Irgens LM, Haagaas I, Skranes JS, Meberg AE, Vik T. Cerebral
palsy in Norway: prevalence, subtypes and severity. Eur J Paediatr Neurol.
2008;12(1):413. https://doi.org/10.1016/j.ejpn.2007.05.001.
5. Hollung SJ, Vik T, Wiik R, Bakken IJ, Andersen GL. Completeness and
correctness of cerebral palsy diagnoses in two health registers: implications
for estimating prevalence. Dev MedChild Neurol. 2017;59(4):4026. https://
doi.org/10.1111/dmcn.13341.
6. Altonoby A, Tawfek M, Abdelaziem F, Kilany A. Establish registry of cerebral
palsy in Tanta Egypt. International Journal of Therapies & Rehabilitation
Research. 2017;6(2):1749. https://doi.org/10.5455/ijtrr.000000260.
7. Serdaroglu A, Cansu A, Md SÖ, Tezcan S. Prevalence of cerebral palsy in
Turkish children between the ages of 2 and 16 years. Dev Med Child
Neurol. 2006;48(6):4136. https://doi.org/10.1111/j.1469-8749.2006.tb01288.x.
8. The Third Millenium Development Goals Report. Republic of Moldova,
2013. www.undp.md/publications/Millenium_ROM_small.pdf
9. CIA Fact book. Retrieved from: https://www.cia.gov/library/publications/the-
world-factbook/geos/md.html, August 2018.
10. Surveillance of cerebral palsy in Europe: a collaboration of cerebral palsy
surveys and registers. Surveillance of Cerebral Palsy in Europe (SCPE). Dev
Med Child Neurol. 2000;42(12):81624.
11. World Health Organisation. International statistical classification of diseases
and related health problems, 10
th
revision (ICD-10). Geneva: World Health
Organisation; 1992.
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. Devel Med Child Neurol. 1997;39(4):21423.
https://doi.org/10.1111/j.1469-8749.1997.tb07414.x.
13. Beckung E, Hagberg G. Neuroimpairments, activity limitations, and
participation restrictions in children with cerebral palsy. Developmental
Medicine & Child Neurology. 2002;44(5):30916. https://doi.org/10.1111/j.
1469-8749.2002.tb00816.x.
14. Simeonsson RJ, Rosenthal SL. Psychological & Developmental Assessment.
Development Assesment of young children (DAYC evaluation), vol. 386.
New York: Guilford Press; 2001. ISBN 1-57230-645-9
15. Cans C, Dolk H, Platt MJ, Colver A, Prasauskiene A, RÄGeloh-Mann IK.
Recommendations from the SCPE collaborative group for defining and
classifying cerebral palsy. Dev Med Child Neurol. 2007;49:358. https://doi.
org/10.1111/j.1469-8749.2007.tb12626.x.
16. Himpens E, Van den Broeck C, Oostra A, Calders P, Vanhaesebrouck P.
Prevalence, type, distribution, and severity of cerebral palsy in relation to
gestational age: a meta-analytic review. Dev Med Child Neurol. 2008;50(5):
33440. https://doi.org/10.1111/j.1469-8749.2008.02047.x.
17. Sellier E, Horber V, KrÄGeloh-Mann I, De La Cruz J, Cans C, On behalf of the,
S. C. Interrater reliability study of cerebral palsy diagnosis, neurological
subtype, and gross motor function. Dev Med Child Neurol. 2012;54(9):815
21. https://doi.org/10.1111/j.1469-8749.2012.04359.x.
18. Howard, J., Soo, B., Graham, H. K., Boyd, R. N., Reid, S., Lanigan, A., . . .
Reddihough, D. S. (2005). Cerebral palsy in Victoria: motor types,
topography and gross motor function. J Paediatr Child Health, 41(910),
479483. doi:https://doi.org/10.1111/j.1440-1754.2005.00687.x.
19. 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):41723. https://doi.org/10.1017/S0012162206000922.
20. Wichers M, Hilberink S, Roebroeck ME, Stam HJ, van Nieuwenhuizen O.
Motor impairments and activity limitations in children with spastic cerebral
palsy: a Dutch population-based study. J Rehabil Med. 2009;41(5):36774.
https://doi.org/10.2340/16501977-0339.
21. Andersen G, Mjøen TR, Vik T. Prevalence of speech problems and the use of
augmentative and alternative communication in children with cerebral palsy: a
registry-based study in Norway. Perspectives on Augmentative and Alternative
Communication. 2010;19(1):12. https://doi.org/10.1044/aac19.1.12.
22. National Bureau of Statistics of the Republic of Moldova, statbank.statistica.
md, Annu Rep 2009, 2010.
23. Ala Curteanu, Characteristics of physical development of premature
newborns in the first 2 years of life Retrieved from: https://ibn.idsi.md/sites/
default/files/imag_file/Particularitatile%20dezvoltarii%20fizice%20a%20nou_
nascutilor.pdf
24. Meberg A, Broch H. Etiology of cerebral palsy. In: Journal of Perinatal
Medicine, vol. Vol. 32, pp. 434; 2004.
25. Smithers-Sheedy H. Declining prevalence of cerebral palsy in Europe: good
news? Dev Med Child Neurol. 2016;58(1):144. https://doi.org/10.1111/
dmcn.12885.
26. Jystad, K. P., Strand, K. M., Bjellmo, S., Lydersen, S., Klungsöyr, K., Stoknes, M., .
. . Vik, T. (2017). Congenital anomalies and the severity of impairments for
cerebral palsy. Dev Med Child Neurol, 59(11), 11741180. doi:https://doi.org/
10.1111/dmcn.13552.
27. Khandaker, G., Van Bang, N., Dũng, T. Q., Giang, N. T. H., Chau, C. M., Van
Anh, N. T., . . . Elliott, E. J. (2017). Protocol for hospital based-surveillance of
cerebral palsy (CP) in Hanoi using the Paediatric active enhanced disease
surveillance mechanism (PAEDS-Vietnam): a study towards developing
hospital-based disease surveillance in Vietnam. BMJ Open, 7(11). e017742
doi:https://doi.org/10.1136/bmjopen-2017-017742.
Gincota Bufteac et al. BMC Pediatrics (2018) 18:332 Page 9 of 9
Content courtesy of Springer Nature, terms of use apply. Rights reserved.
1.
2.
3.
4.
5.
6.
Terms and Conditions
Springer Nature journal content, brought to you courtesy of Springer Nature Customer Service Center GmbH (“Springer Nature”).
Springer Nature supports a reasonable amount of sharing of research papers by authors, subscribers and authorised users (“Users”), for small-
scale personal, non-commercial use provided that all copyright, trade and service marks and other proprietary notices are maintained. By
accessing, sharing, receiving or otherwise using the Springer Nature journal content you agree to these terms of use (“Terms”). For these
purposes, Springer Nature considers academic use (by researchers and students) to be non-commercial.
These Terms are supplementary and will apply in addition to any applicable website terms and conditions, a relevant site licence or a personal
subscription. These Terms will prevail over any conflict or ambiguity with regards to the relevant terms, a site licence or a personal subscription
(to the extent of the conflict or ambiguity only). For Creative Commons-licensed articles, the terms of the Creative Commons license used will
apply.
We collect and use personal data to provide access to the Springer Nature journal content. We may also use these personal data internally within
ResearchGate and Springer Nature and as agreed share it, in an anonymised way, for purposes of tracking, analysis and reporting. We will not
otherwise disclose your personal data outside the ResearchGate or the Springer Nature group of companies unless we have your permission as
detailed in the Privacy Policy.
While Users may use the Springer Nature journal content for small scale, personal non-commercial use, it is important to note that Users may
not:
use such content for the purpose of providing other users with access on a regular or large scale basis or as a means to circumvent access
control;
use such content where to do so would be considered a criminal or statutory offence in any jurisdiction, or gives rise to civil liability, or is
otherwise unlawful;
falsely or misleadingly imply or suggest endorsement, approval , sponsorship, or association unless explicitly agreed to by Springer Nature in
writing;
use bots or other automated methods to access the content or redirect messages
override any security feature or exclusionary protocol; or
share the content in order to create substitute for Springer Nature products or services or a systematic database of Springer Nature journal
content.
In line with the restriction against commercial use, Springer Nature does not permit the creation of a product or service that creates revenue,
royalties, rent or income from our content or its inclusion as part of a paid for service or for other commercial gain. Springer Nature journal
content cannot be used for inter-library loans and librarians may not upload Springer Nature journal content on a large scale into their, or any
other, institutional repository.
These terms of use are reviewed regularly and may be amended at any time. Springer Nature is not obligated to publish any information or
content on this website and may remove it or features or functionality at our sole discretion, at any time with or without notice. Springer Nature
may revoke this licence to you at any time and remove access to any copies of the Springer Nature journal content which have been saved.
To the fullest extent permitted by law, Springer Nature makes no warranties, representations or guarantees to Users, either express or implied
with respect to the Springer nature journal content and all parties disclaim and waive any implied warranties or warranties imposed by law,
including merchantability or fitness for any particular purpose.
Please note that these rights do not automatically extend to content, data or other material published by Springer Nature that may be licensed
from third parties.
If you would like to use or distribute our Springer Nature journal content to a wider audience or on a regular basis or in any other manner not
expressly permitted by these Terms, please contact Springer Nature at
onlineservice@springernature.com
... These limited financial resources are a likely explanation for the high maternal mortality rate of 23 deaths per 100,000 live births-five to six times higher than in the Nordic countries, and of the infant mortality rate of 12 deaths per 1000 live births. The prevalence of CP and distribution of CP subtypes, motor impairments and associated problems were unknown till 2018 [1]. ...
... The first study on prevalence, subtypes, severity and associated impairments was conducted by the authors of the present article [1]. As a next step we wanted to look into the potential impact on impairments of the early intervention and follow-up programs among children with CP [3]. ...
... The recruitment area of the present study covered both rural and urban localities in Moldova. To perform this study, we first conducted a cohort study that allowed us to describe the characteristics of total births for two years (2009)(2010): total number of births, neonatal mortality, gestational age, multiple gestations and birth presentation [1]. ...
Article
Full-text available
Background and Objectives: This is the first study assessing risk factors for cerebral palsy (CP) among children born in Moldova. The aim of this study was to identify and describe risk factors for cerebral palsy (CP) among children born in Moldova, which is one of the low-middle income countries in Europe. Materials and Methods: We identified 351 children with CP born during 2009 and 2010 in Moldova. Detailed information on 417 children without CP served as a reference group. Logistic regression analyses were applied to the calculate crude and adjusted odds ratios (OR) for CP with 95% confidence intervals (CI) in addition to attributable fraction (AF). Results: Among children with CP (40.5% girls), 26% had spastic unilateral, 54% bilateral, 13% dyskinetic, 5% ataxic and 2% unclassified CP. Significant risk factors for CP included maternal alcohol consumption during pregnancy (OR 1.7, p = 0.002), maternal hypertension (OR 2.0, p < 0.001), children born to mothers from the rural areas (OR 1.6, p < 0.001), maternal age ≥35 years (OR 0.6, p = 0.018), maternal epilepsy (OR 4.3, p < 0.001), breech delivery (OR 3.1, p = 0.001), home births (OR 6.3, p = 0.001), umbilical cord around neck (OR 2.2, p < 0.001), AVD (OR 3.1, p < 0.001), male gender (OR 1.3, p < 0.001), SGA (OR 1.3, p = 0.027), multiple gestations (OR 1.7, p < 0.001) and hyperbilirubinemia (OR 4.5, p < 0.001). Multivariable analyses showed that the AF of CP was 64% for rural residence (OR 2.8, p = 0.002), 87% for home birth (7.6, p = 0.005), 79% for pre-labor rupture of membrane (OR 4.9, p = 0.001), 66% for breech delivery (OR 2.9, p = 0.002) and 81% for hyperbilirubinemia (OR 5.4, p < 0.001). Conclusions: A combination of factors related to the mother, the delivery and the child were risk factors for CP in Moldova, many of them possibly avoidable. Improved pregnancy and maternity care would potentially reduce the risk of CP. A national CP registry in Moldova is suggested as an opportunity to follow up on these findings.
... In this way, we succeeded in covering approximately 95% of the population. In the rst article on CP in Moldova from 2018, we described a cohort based on 75% population coverage, with subtypes, severity and prevalence of CP diagnosed in children [14]. ...
... Orphanages and habilitation centres in Moldova also provided summary and detailed perinatal and clinical data. SCPE criteria for the diagnosis of CP were applied based on detailed neurological descriptions in the medical records [2,14]. Only those children with CP with complete perinatal information were included in the present study. ...
... The CP diagnosis was classi ed in accordance with SCPE recommendations by the rst author, based on detailed neurological descriptions from the children's medical records [2]. However, the cohort for the present study is small, only two birth years Comparison with other studies Our rst study from 2018, on the prevalence and severity of CP in Moldova in comparison to industrial developed countries in Europe, showed an almost two-fold increase in the number of children with severe forms of CP and associated impairments [14]. The present manuscript is a follow-up study to examine in depth the potential risk factors for CP from three presented groups of variables. ...
Preprint
Full-text available
Background This is the first study assessing risk factors for cerebral palsy (CP) among children born in Moldova.Methods We identified 351 children with CP born during 2009 and 2010 in state hospitals, maternity wards, orphanages and rehabilitation centres in Moldova. Detailed information on 417 children without CP (of the total number of 81,277 children) served as a reference group. Logistic regression analyses were applied to calculate crude and adjusted odds ratios (OR) with 95% confidence intervals (CI) in addition to attributable risk (AR).ResultsAmong children with CP (40.5% girls), 26% had unilateral, 54% bilateral, 13% dyskinetic, and 5% ataxic CP, with 2% unclassified. Almost three quarters of children with CP were from rural areas and 101 (29%) were prematurely born.Significant risk factors for CP included low Apgar score at five minutes (OR 6.187, 95% CI 2.059–18.59), hyperbilirubinemia (OR 6.3, 95% CI 4.1–9.6), breech delivery (OR 3.5, 95% CI 2–6.01), assisted vaginal delivery (OR 47, 95% CI 11–194), birth out of maternity (OR 6.591, 95% CI 1.904–22.81), low education level (OR 3.889, 95% CI 2.8–5.248), alcohol consumption (OR 1.903, 95% CI 1.269–2.885) and hypertension (OR 2.264, 95% CI 1.5–5.417).For singletons born at term, significant risk factors were low Apgar score (OR 2.115, 95% CI 1.22–3.808), hyperbilirubinemia (OR 5.854, 95% CI 3.648–9.393), breech delivery (OR 6.48, 95% CI 3.1–13.7), fundal pressure (OR 3.268, 95% CI 1.398–7.642), maternal hypertension (OR 2.248, 95% CI 1.55–3.997) and alcohol consumption (OR 2.105, 95% CI 1.315–3.369).Hyperbilirubinemia, Apgar score (0–3), birth out of maternity and breech delivery had the highest AR for CP both in the total and term singleton groups. Chronic diseases among mothers also had a high AR for CP.ConclusionsA combination of factors related to the child, the delivery and the mother were risk factors for CP among children born in Moldova, many of them possibly avoidable. The results may indicate a need for better maternal and perinatal childcare. A national CP register in Moldova is recommended as an opportunity to follow up these findings.
... Variation in birth prevalence exists in highincome countries (1.4 to 2.5 affected children per 1000 live births) and is higher again in low-to middle-income countries. [1][2][3][4][5] Cerebral palsy occurs because of a lesion or maldevelopment in the developing brain. Often, the full causal pathway is unclear. ...
... 10 Comprehensive systematic searches, using the Cochrane method, were conducted for the 9 topics within the target age range using Patient, Intervention, Comparison, and Outcome (PICO) 11 searches (eTable 1 in the Supplement). The inclusion criteria specified (1) gathering existing systematic reviews answer-ing the PICO questions; (2) when systematic reviews were not available, conducting a new systematic review to identify randomized clinical trials (RCTs) or the best available lower levels of evidence when systematic reviews and RCTs were not available 12 ; or (3) when no or limited data on CP in children aged 0 to 2 years were published, conducting searches to identify RCTs in patients with other neurodevelopmental disabilities or older children with CP (Table). ...
Article
Full-text available
Importance Cerebral palsy (CP) is the most common childhood physical disability. Early intervention for children younger than 2 years with or at risk of CP is critical. Now that an evidence-based guideline for early accurate diagnosis of CP exists, there is a need to summarize effective, CP-specific early intervention and conduct new trials that harness plasticity to improve function and increase participation. Our recommendations apply primarily to children at high risk of CP or with a diagnosis of CP, aged 0 to 2 years. Objective To systematically review the best available evidence about CP-specific early interventions across 9 domains promoting motor function, cognitive skills, communication, eating and drinking, vision, sleep, managing muscle tone, musculoskeletal health, and parental support. Evidence Review The literature was systematically searched for the best available evidence for intervention for children aged 0 to 2 years at high risk of or with CP. Databases included CINAHL, Cochrane, Embase, MEDLINE, PsycInfo, and Scopus. Systematic reviews and randomized clinical trials (RCTs) were appraised by A Measurement Tool to Assess Systematic Reviews (AMSTAR) or Cochrane Risk of Bias tools. Recommendations were formed using the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) framework and reported according to the Appraisal of Guidelines, Research, and Evaluation (AGREE) II instrument. Findings Sixteen systematic reviews and 27 RCTs met inclusion criteria. Quality varied. Three best-practice principles were supported for the 9 domains: (1) immediate referral for intervention after a diagnosis of high risk of CP, (2) building parental capacity for attachment, and (3) parental goal-setting at the commencement of intervention. Twenty-eight recommendations (24 for and 4 against) specific to the 9 domains are supported with key evidence: motor function (4 recommendations), cognitive skills (2), communication (7), eating and drinking (2), vision (4), sleep (7), tone (1), musculoskeletal health (2), and parent support (5). Conclusions and Relevance When a child meets the criteria of high risk of CP, intervention should start as soon as possible. Parents want an early diagnosis and treatment and support implementation as soon as possible. Early intervention builds on a critical developmental time for plasticity of developing systems. Referrals for intervention across the 9 domains should be specific as per recommendations in this guideline.
... With regards to comorbidities, we reported similar findings in epilepsy, visual impairment, cognitive impairment and hip displacements as most other registries. 4,13,23,24,31,32 In terms of gross motor function, our Registry reported higher proportions (45%) of participants with severe impairments (GMFCS IV-V, 25-28% in established CP registries). 4,24 This may be explained by our Registry being hospital-and centrebased, as compared to other community registries. ...
... 29 In our Registry, dyskinetic CP represent a larger proportion (27.8% overall) as compared to 7-12% in Europe and Australia. 4,30,31 This may be related to specialist recognition of dyskinesia in our hospitalbased cohort. Separately, due to a rigorous national ...
Article
Full-text available
Introduction: A voluntary cerebral palsy (CP) registry was established in 2017 to describe the clinical characteristics and functional outcomes of CP in Singapore. Methods: People with CP born after 1994 were recruited through KK Women's and Children's Hospital, National University Hospital and Cerebral Palsy Alliance Singapore. Patient-reported basic demographics, service utilisation and quality of life measures were collected with standardised questionnaires. Clinical information was obtained through hospital medical records. Results: Between 1 September 2017 and 31 March 2020, 151 participants were recruited. A majority (n=135, 89%) acquired CP in the pre/perinatal period, where prematurity (n=102, 76%) and the need for emergency caesarean section (n=68, 50%) were leading risk factors. Sixteen (11%) of the total participants had post-neonatally acquired CP. For predominant CP motor types, 109 (72%) had a spastic motor type; 32% with spastic mono/hemiplegia, 41% diplegia, 6% triplegia and 21% quadriplegia. The remaining (42, 27.8%) had dyskinetic CP. Sixty-eight (45.0%) participants suffered significant functional impairment (Gross Motor Functional Classification System levels IV-V). Most participants (n=102, 67.5%) required frequent medical follow-up (≥4 times a year). Conclusion: Optimisation of pre- and perinatal care to prevent and manage prematurity could reduce the burden of CP and their overall healthcare utilisation.
... Gross motor function was classified according to the Gross Motor Function Classification System (GMFCS) [16]. As the data were collected from descriptions in medical records of variable quality, the children's gross motor function was dichotomised into walking (GMFCS levels I, II and III), and non-walking (GMFCS levels IV and V) children, as proposed by Andersen et al. [17] and used in a previous study by Gincota et al. [18]. ...
... Fine motor function was classified according to the Bimanual Fine Motor Function (BFMF) scale [19] and was described by speech therapists as free text in medical records, as the development of communication at an early stage is closely related to use of hands, and dichotomised into BFMF level I-III, and BFMF levels IV and V as proposed by Andersen et al. [17] and used in a previous study by Gincota et al. [18]. ...
Article
Full-text available
Aim: To study whether early intervention services (EI) and a follow-up program (FU) influence outcomes of children with cerebral palsy (CP) in Moldova. Methods: Records from 351 children with CP in Moldova born during 2009 and 2010 were retrieved from hospital and orphanage archives between 1 July 2016 and 30 September 2017. We investigated the proportion enrolled in EI and FU at the Early Intervention Centre Voinicel and at the Institute of Mother and Child in 2009-2012. Logistic regression analyses were applied to calculate crude and adjusted odds ratios (OR) with 95% confidence intervals (CI) for outcomes in children enrolled and not enrolled. Results: Among all children with CP, 166 (47%) were enrolled in EI and FU. Of the 51 children born extremely preterm (gestational age ≤ 31 weeks), 46 (90%) were enrolled, compared to 97 (39%) of the 250 children born at term. Among 110 non-walking children with CP, 82 (74%) were enrolled into EI and FU, compared to 84 (35%) of 241 able to walk. There was no difference in outcomes of cognition, communication, vision and hearing impairments between those enrolled or not enrolled in EI and FU. However, the subgroup analyses showed that the risk of contractures was 11 times higher among non-walking children who were not enrolled in EI and FU programs (OR = 10.931, 95% CI 2.328-51.328, p = 0.002). Conclusion: In Moldova, EI and FU seem to be offered mostly to extremely preterm and non-walking children with CP. The results indicate a decreased risk for contractures in these children.
... Cerebral palsy (CP) is described as "a group of permanent disorders of the development of movement and posture, causing activity limitations that are attributed to non-progressive disturbances that occurred in the developing foetal or infant brain" (Rosenbaum et al., 2007, p. 9). Using advances in medical technology and methodologies, population based studies from around the world report an estimated prevalence of CP to be 1.5-4 per 1000 live births (Maenner et al., 2016;Smithers-Sheedy et al., 2016;Stavsky et al., 2017;Van Naarden Braun et al., 2016), each study reporting different motor types of CP to be the most prevalent among their cohort (Bufteac, Andersen, Torstein, & Jahnsen, 2018;Durkin et al., 2015;El-Tallawy et al., 2014;Kakooza-Mwesige, Forssberg, Eliasson, & Tumwine, 2015;Sigurdardottir, Thorkelsson, Halldorsdottir, Thorarensen, & Vik, 2009;Singhi & Saini, 2013). The commonly used classifications to characterise CP are: a) Gross Motor Function Classification system (GMFCS) ( Palisano et al., 1997) that divides physical disability based on sitting, walking and wheeled mobility; b) Manual Ability Classification system (MACS) which categorises manual performance ( Eliasson et al., 2006) and c) Communication Function Classification System (CFCS) which classifies the communication skills of a child with CP with both familiar and unfamiliar partners in different environments (Hidecker et al., 2011). ...
... There is a well-known relationship between prematurity and CP, however, only a recently performed Moldavian study further classified VI according to GA in children with CP (30). In our study, extremely preterm infants with bilateral spastic CP showed the highest prevalence of severe VI (53.8%). ...
Article
Aim: To evaluate visual impairment (VI) in children with cerebral palsy (CP). Methods: This population-based study included 419 children from the Surveillance of Cerebral Palsy in Europe (SCPE) C28 RCP-HR - Register of Cerebral Palsy of Croatia born 2003-2008. Vision in children with CP (according to SCPE) was classified as normal or impaired, with the subcategory of severe VI. The proportion of children with VI was assessed in groups with different CP type/subtype, gross and fine motor function, and gestational age (GA). Results: A total of 266 children had some degree of VI (266/400; 66.5%), 134 had normal vision, and data on VI were unknown for 19 children. Severe VI was present in 44 children (44/400; 11%). The proportion of children with VI and severe VI increased with the Gross Motor Function Classification System and Bimanual Fine Motor Function levels. Children with bilateral spastic CP had the highest frequency of severe VI (14.9%). The percentage of severe VI in children with bilateral spastic CP was 53.8% in the group born <28 weeks of GA, 13.3% in the group born 28-31 weeks of GA, 11.1% in the group born 32-36 weeks of GA, and 24.4% in the group born >36 weeks of GA (λ2=4.95; df=6; P<0.001). Conclusion: Children with CP have a high prevalence of VI and severe VI, which is increasing with the level of motor impairment. Severe VI is significantly more common in children with bilateral spastic CP, especially among extremely premature infants.
Article
Full-text available
Objective To describe the frequency, motor phenotype, clinical patterns and functional consequences of dystonia in patients with cerebral palsy (CP) in the setting of periventricular leukomalacia. Methods Retrospective analysis of a cohort of 31 patients with CP and periventricular leukomalacia. Gross Motor Function Classification System (GMFCS) and Manual Ability Classification System (MACS) were used to classify functional ability. Spasticity was rated using the Modified Ashworth Scale. Presence of dystonia was assessed by reviewing video recordings, and its severity by using the Burke–Fahn–Marsden Dystonia Rating Scale. Results All patients showed evidence of dystonia involving upper and/or lower limbs, neck, trunk, mouth and eyes in order of frequency. In 29% of patients dystonia involved only the limbs and in 71% it was multifocal. Dystonia severity ranged from slight to severe. Severity and distribution of dystonia did not correlate with gender, age, weeks of gestation or duration of neonatal unit stay. GMFCS and MACS correlated with dystonia but not with spasticity. Conclusions Severity of dystonia, but not spasticity is associated with the severity of motor functional disability in CP patients with periventricular leukomalacia and demonstrates the key role of dystonia in the motor function of these patients.
Article
Objective: To evaluate the most typical target muscles and dosages for the first and repeated botulinum toxin A (BTA) injections in cerebral palsy (CP) patients with severe motor deficit - GMFCS IV-V. Material and methods: A retrospective analysis of 677 protocols of the first and repeated Abobotulinumtoxin A (AboA) injections in 333 patients with CP GMFCS IV and V, aged 1 to 18 years, was carried out. Results: Ninety-seven percent of patients received multilevel injections. In the lower extremities the most typical target muscles were: m.gracilis - 221 (66.4%) patients, hip adductors - 164 (49.2%), medial hamstring - 144 (43.2%). In the upper extremities the most typical muscles were: m.pronator teres - 237 (71.2%) patients, m.biceps brachii+m.brachialis - 197 (59.2%). The total dosages of AboA and dosages for every target muscle were calculated. Several patients required high dosages (more than 30 U/kg of AboA). Higher dosages per kg were used in younger children and for repeated injections. The age-related evolution of spastic patterns was described. Adverse events were observed in 36 cases (5.3% of all injections). Conclusion: The majority of patients with GMFCS IV-V required multilevel BTA injections in high dosages, especially in young age. Described selection of target muscles and dosages of AboA could be taken into account as a practical experience and reference for the BTA therapy in GMFCS IV-V patients.
Article
Full-text available
Introduction The epidemiology, pathogenesis, management and outcomes of cerebral palsy (CP) in low-income and middle-income countries including Vietnam are unknown because of the lack of mechanisms for standardised collection of data. In this paper, we outline the protocol for developing a hospital-based surveillance system modelled on the Paediatric Active Enhanced Disease Surveillance (PAEDS) system in Australia. Using PAEDS-Vietnam we will define the aetiology, motor function and its severity, associated impairments, and nutritional and rehabilitation status of children with CP in Hanoi, Vietnam. These essential baseline data will inform future health service planning, health professional education and training, and family support. Methods and analysis This is a hospital-based prospective surveillance of children with CP presenting to the rehabilitation, neurology and general paediatric services at the National Children’s Hospital and St Paul Hospital in Hanoi. We will use active, prospective daily case-finding for all children with CP aged <18 years who are hospitalised or present to outpatient departments. Following parental consent, data will be collected using a modified version of the Australian Cerebral Palsy Register questionnaire. The data collection form has been developed in consultation with local and international experts and translated into Vietnamese. Information collected will include demographics, maternal health and birth history, type and severity of CP, known risk factors for CP, and nutrition, immunisation, education and rehabilitation status. Ethics and dissemination This study was approved by the Hanoi Medical University Institutional Review Board (decision no 1722) and The University of Sydney Human Research Ethics Committee (approval no 2016/456). Establishment of PAEDS-Vietnam will enable hospital-based surveillance of CP for the first time in Vietnam. It will identify preventable causes of CP, patient needs and service gaps, and facilitate early diagnosis and intervention. Study findings will be disseminated through local and international conferences and peer-reviewed publications.
Article
Full-text available
Aim: To assess completeness and correctness of cerebral palsy (CP) diagnoses in the Cerebral Palsy Register of Norway (CPRN) and the Norwegian Patient Register (NPR), and to estimate CP prevalence. Method: Among 747 883 Norwegian residents born from 1996 to 2007, 2231 had a diagnosis of CP in the NPR while 1441 were registered in the CPRN. Children registered in the CPRN were considered to have a valid CP diagnosis. For those with a diagnosis of CP only in the NPR, two paediatricians reviewed the hospital records. The prevalence rate of CP with 95% confidence intervals (CI) was calculated on the basis of the combined data sets. Results: One thousand three hundred and ninety-eight children were registered with a diagnosis of CP in both registers, 43 children were only registered in the CPRN, and 824 only in the NPR. The review of hospital records revealed that 464 (59.5%) had CP. Thus, the NPR was 98% complete, and for 86% the diagnosis was correct. The completeness of the CPRN was 76%, while the diagnosis was considered correct for all children (100%). The resulting prevalence of CP was 2.5 (95% CI 2.4-2.7) per 1000. Interpretation: To gain accurate estimates of prevalence rates of CP, it is essential to combine data sources and to validate register data.
Article
Full-text available
Cerebral palsy (CP) is the most frequent cause of motor handicap. The present door-to-door survey was conducted in Al-Quseir City, Egypt, to investigate the epidemiology of CP. All inhabitants were screened by three neurologists. Medical and neurological examinations were performed for all residents and suspected cases of CP were confirmed by meticulous neurological assessment, brain magnetic resonance imaging, electroencephalography, and testing with the Stanford-Binet Intelligence Scale. Forty-six of 12,788 children aged ≤18 years were found to have CP, yielding a childhood prevalence of 3.6 (95% confidence interval 1.48-2.59) per 1,000 live births. Five adults (aged 19-40 years) among 13,056 inhabitants had CP, giving an adult prevalence of 0.4 (95% confidence interval 0.04-0.72) per 1,000. The risk factors for CP identified in this study were premature birth, low birth weight, neonatal jaundice, neonatal seizures, and recurrent abortion in mothers of children with CP.
Article
Full-text available
This study describes the prevalence of speech problems and the use of augmentative and alternative communication (AAC) in children with cerebral palsy (CP) in Norway. Information on the communicative abilities of 564 children with CP born 1996–2003, recorded in the Norwegian CP Registry, was collected. A total of 270 children (48%) had normal speech, 90 (16%) had slightly indistinct speech, 52 (9%) had indistinct speech, 35 (6%) had very indistinct speech, 110 children (19%) had no speech, and 7 (1%) were unknown. Speech problems were most common in children with dyskinetic CP (92 %), in children with the most severe gross motor function impairments and among children being totally dependent on assistance in feeding or tube-fed children. A higher proportion of children born at term had speech problems when compared with children born before 32 weeks of gestational age 32 (p < 0.001). Among the 197 children with speech problems only, 106 (54%) used AAC in some form. Approximately 20% of children had no verbal speech, whereas ~15% had significant speech problems. Among children with either significant speech problems or no speech, only 54% used AAC in any form.
Article
Aim: To study the prevalence of congenital anomalies among children with cerebral palsy (CP) born at term or late preterm, and if CP subtypes and clinical manifestations differ between children with and without congenital anomalies. Method: This was a cross-sectional study using data from the Cerebral Palsy Register of Norway and the Medical Birth Registry of Norway. All children with congenital CP born at and later than 34 weeks' gestation in Norway from 1999 to 2009 were included. Anomalies were classified according to the European Surveillance of Congenital Anomalies classification guidelines. Groups were compared using Fisher's exact test, Kruskal-Wallis test, and the Mann-Whitney U test. Results: Among 685 children with CP, 169 (25%) had a congenital anomaly; 125 within the central nervous system. Spastic bilateral CP was more prevalent in children with anomalies (42%) than in children without (34%; p=0.011). Children with anomalies less frequently had low Apgar scores (p<0.001), but more often had severe limitations in gross- and fine-motor function, speech impairments, epilepsy, severe vision, and hearing impairments than children without anomalies (p<0.03). Interpretation: Although children with CP and anomalies had low Apgar scores less frequently, they had more severe limitations in motor function and more associated problems than children with CP without anomalies. What this paper adds: One in four children with cerebral palsy (CP) born at term or late preterm has a congenital anomaly. The added value of neuroimaging to detect central nervous system anomalies in children with CP. Children with anomalies have more severe motor impairments. More severe clinical manifestations are not explained by perinatal complications as indicated by low Apgar scores.
Article
This commentary is on the original article by Sellier et al.
Article
To evaluate the interrater reliability of the inclusion in registries and classification of children with cerebral palsy (CP). Two studies were conducted. In study 1, 12 paediatricians from 11 countries viewed video sequences of 12 children with or without CP (nine males, three females; median age 6y; range 2-16). In study 2, 19 professionals from eight countries participated in an online exercise. They had to classify the same children but based on written vignettes. All participants had to evaluate whether the child had CP, the neurological subtype (Surveillance of Cerebral Palsy in Europe classification system), and gross motor function level (Gross Motor Function Classification System [GMFCS]). Kappa (κ) coefficients were calculated for categorical variables and intraclass correlation coefficients (ICCs) for ordinal data. Reliability was excellent in assessing whether or not a child had CP in study 1 (κ=1.00) and substantial in study 2 (κ=0.73); 95% confidence interval [CI] 0.58-0.87). For the neurological subtype, overall κ between paediatricians was 0.85 (95% CI 0.68-0.98), with full agreement observed for eight children. In study 2, overall κ was 0.78 (95% CI 0.61-0.91) with full agreement seen for five children. For the GMFCS, the ICC was 0.88 (95% CI 0.78-0.95) in study 1 and 0.80 (95% CI 0.64-0.91) in study 2. Reliability was excellent for all characteristics classified by paediatricians viewing the videos and substantial for professionals reading vignettes.
Article
The aim of this review is to determine the relationship between gestational age (GA) and prevalence, type, distribution, and severity of cerebral palsy (CP). Epidemiological studies with cohorts expressed by GA were assessed. A comprehensive meta-analysis and meta-regression was performed on four fetal age categories. Studies of children with CP as a target population were added. Twenty-six articles met the inclusion criteria. The prevalence of CP decreases significantly with increasing GA category: 14.6% at 22 to 27 weeks' gestation, 6.2% at 28 to 31 weeks, 0.7% at 32 to 36 weeks, and 0.1% in term infants. Interestingly, a significant decrease in prevalence of CP starts only from a GA of 27 weeks onwards. In preterm infants, spastic CP is predominant. In term infants, the non-spastic form of CP is more prevalent than in preterm infants. Bilateral spastic CP is most prevalent in both preterm and term infants. However, the proportion of unilateral spastic CP in term infants is substantial. No relationship could be detected between severity of CP and GA. There is a strong need for an international, well-described, and generally accepted classification system for subtypes and severity of CP.