1.2.1

Type of study

Transversal descriptive study based on a self-report questionnaire.

1.2.2

Sample

Between February 2010 and June 2011, 450 questionnaires and prepaid envelopes were sent to 40 physicians with case load of children with CP in the four departments of Brittany (neuropediatricians, physiatrists, pediatric surgeons, doctors from the Medico-Education Institute, Institute of Motor Education, Center of early medico-social assistance, Education Department and Specialized home care). The doctors in this network distributed the questionnaire to the parents. Phone call and email reminders were regularly sent to the physicians during this period.

1.2.3

The questionnaire

The aim of the questionnaire was to define current medical management of children with CP by capturing a specific instant of their treatment. The questions asked to the parents concerned medical and paramedical involvement at the time that the questionnaire was filled in: oral medication (the prescription was copied on to the questionnaire), botulinum toxin injections (within the last 6 to 12 months) and the site(s) of the limb(s) injected, baclofen pumps, mobility and postural aids, medical input and rehabilitation (frequency). The questionnaire was based on the recommendations of McColl et al. and met the following criteria:

• •
  

  each questionnaire was given to the parents along with an information letter which explain the aims of the questionnaire and guaranteed freedom of reply, confidentiality and anonymity;

  
  
• •
  

  it was short enough to take less than 15 minutes to fill out and the language used was simple. Technical terms were explained by photos (for example, walking frame, adapted seating, etc.);

  
  
• •
  

  the presentation of the questionnaire was as light as possible, using tables with mostly tick boxes. If the parents wanted to make further comments, there was a free area at the end of the questionnaire;

  
  
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  the first page was for the doctors in order to confirm the diagnosis of CP (the only inclusion criterion for the study), the type of CP (unilateral spastic, bilateral spastic, dyskinetic, ataxic or other), the limbs involved and cognitive function (no, slight or severe loss of function). In order to be able to link the first page to the rest of the questionnaire, a number was attributed to each questionnaire.

1.2.4

The GMFCS classification

The parents were asked to rate their children according to the GMFCS. Two different simplified GMFCS texts were used for children aged 4–6 and 6–18. This procedure has already been shown to be reliable for these age groups . Both the texts were translated by a bilangual English-French speaking physiotherapist. The text used for children aged 2–4 years old was the complete GMFCS which had already been translated into French . In the questionnaire, level V of the classification relating to the children with the most reduced motor function was positioned at the start of the questionnaire, before the description of the motor level, so that the parents did not have to read all the activities which their child could not carry out. The other levels were presented in decreasing order.

1.2.5

Statistical analysis

Data were input using EPIDATA ^® software and statistical analysis was carried out using SAS 9.1 ^® . The different characteristics of the sample were described according to medical and paramedical input. A univariate test was carried out to evaluate if and how the degree of input changed depending on the GMFCS score. The variables of interest were either qualitative variables (physiotherapy [yes or no], etc.) or quantitative (number of consultations, number of assistive devices, etc.). A Cochran Armitage test was used to evaluate tendencies between qualitative management variables and the GMFCS level. A Spearman’s correlation test was used to evaluate the relationship between the quantitative management variables. P < 0.05 was considered significant in each case.

1.3.1

Population

One hundred and thirty-three of the 450 questionnaires sent to the doctors were returned, giving a response rate of 29.6%. The characteristics of the responders are presented in Table 1 . The majority of responders were mothers (82.7%). The sex-ratio was 1.8 boys to 1 girl and the mean age was 8.4 years. 33.8% of the children were between 2 and 6 years old, 34.6% were between 6 and 12 and 29.3% were between 12 and 18 years old. Thirty percent of the children had unilateral spastic CP, 51.7% had bilateral spastic CP, 17.5% were dyskinetic and 0.8% were ataxic. 40.6% of the children were level I on the GMFCS, 20.3% were level II, 12.03% were level III, 13.53% were level IV and 13.53% were level V. 28.5% had a diagnosed cognitive deficit (21% mild, 7.5% severe).

1.3.2

Drug treatments

The frequencies of drug treatments are presented in Table 2 . 60.9% of children had no oral medication. The four most common types of drug were antiepileptic (17.3%), laxatives (6%), baclofen (4.5%) and analgesics (3.8%). Drug treatment ( P = 0.005), baclofen ( P = 0.005), laxatives ( P = 0.0006) and the number of drugs per child ( P = 0.001) all increased significantly with GMFCS level.

Botulinum toxin injections had been administered to 33.1% of the children within the past 12 months, 21.0% of which were within the previous 6 months. 88.6% had received injections in the lower limbs and 23.2% in the upper limbs. The majority of injections were given to children classed as level II and IV on the GMFCS (50.0% and 44.4%). Two of the 133 children were treated with intrathecal baclofen. One was level IV and the other level V.

1.3.3

Assistive devices

The frequencies of mobility and postural devices are presented in Table 3 . The most common mobility aids were manual wheelchairs (34.6%), followed by posterior rollator walkers (13.3%) and electric wheelchairs (9.0%). Few children used an arrow-walker, stick or anterior walker (1.5 to 3%).

Eighty-five percent of children had at least one orthotic device. The five most common devices were a nighttime ankle-foot orthosis (43.6%), orthopedic insoles (35.4%), seating devices (35.3%), orthopedic shoes (25.6%) and adapted standing frames (24.8%). Upper limb orthoses were uncommon (1.7 to 3.8%).

The number of mobility aids ( P < 0.0001) and orthotic devices ( P = 0.0001) per child increased significantly with the GMFCS level. The type of orthotic and mobility device were also strongly related to the GMFCS level. For example, manual wheelchairs, adapted seating, standing frames, orthopedic shoes and night splints were more common in levels III, IV and V children which corresponds well with the definitions of the GMFCS levels. 62.4% of children wore glasses and this also increased with GMFCS level ( P = 0.01). 3.8% of children wore auditory prostheses.

1.3.4

Paramedical involvement

The frequency of paramedical input is presented in Table 4 . The median number of therapy sessions per week was 3.85 [0.5–11.5]. This number increased with the GMFCS level ( P < 0.0001) from a median of 2 [0.5–6] for level I children, to a median of 5 [2–10] for level V children.

92.6% of level I children and 100% of the other levels had physiotherapy. The median number of sessions per week was 2 [0–5]. 53.4% of children had occupational therapy with a median number of sessions per week of 0.5. 35.3% of children had speech therapy, 32.3% had psychomotor education and 18.8% had vision therapy. 31.6% of the children were followed by a special educator and 30.8% saw a psychologist. The median number of sessions per week of physiotherapy ( P < 0.0001), occupational therapy ( P = 0.0009), speech therapy ( P = 0.064), educator input ( P = 0.002) and psychology ( P = 0.01) increased significantly with GMFCS level.

1.3.5

Medical visits

The frequency of medical visits is presented in Table 5 . Eighty-eight percent of the children were followed by a physiatrist, 75.2% by a general practitioner or pediatrician, 41.3% by a neuropediatrician, and 23.0% by a pediatric surgeon. One child was followed by a psychiatrist. No correlation was found between the different medical practitioners involved and the GMFCS level.

The median number of consultations per year was 5.5 [1–27] with no correlation with GMFCS level. The doctors who were consulted most frequently were the physiatrist and the general practitioner or pediatrician who were consulted a median of twice per year [0–6]. There was a significant correlation between the median number of consultations and the GMFCS level for two specialists: the physiatrist ( P = 0.001) and the surgeon ( P = 0.04).

1.4.1

Drug treatment

This study provides original data regarding drug treatment in a population of children with CP. Forty percent of children had regular medication and the level of medication increased significantly with GMFCS level. Other than antiepileptic drugs, laxatives, baclofen and analgesics were the most frequently prescribed, indicating that constipation, generalized spasticity and pain are a problem, particularly in children with levels III, IV and V of the GMFCS. About one third of the children had received botulinum toxin injections in the year preceding the study and two children received intrathecal baclofen.

In a systematic review on the level of evidence of anti-spastic treatments in CP, Delgado et al. found that :

• •
  

  no oral anti-spastic medication has been correctly evaluated in children (diazepam, dantrolene, baclofen, tizanidine). Particularly, they report that although baclofen is frequently used in clinical practice, there are contradictory studies regarding its effectiveness. The results of the present study show a preference for this molecule among others but with a low level of prescription (4.5% of the children);

  
  
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  botulinum toxin has been shown to be effective in the reduction of spasticity in both the lower and upper limbs but studies regarding the functional benefit of injection are contradictory. Considering this high level of evidence and the fact that 65 to 80% of children with CP have spasticity , the result of 33.1% of children injected within the year appears low. Particularly, the small number of level V children who were injected (16.7%) in this study contrasts with the number of children of other levels who were injected (29.6 to 50%);

  
  
• •
  

  studies regarding the use of intrathecal baclofen do not provide enough evidence to recommend this treatment. The study sample well reflects this lack of consensus.

1.4.2

Assistive devices

Eighty-five percent of the children had at least one orthotic device, the most common was a nighttime ankle-foot orthosis, and over one third had a manual wheelchair. Few children had upper limb orthoses (1.7 to 3.8%). The number of ambulatory aids and orthotic devices and the type of device was highly correlated with the GMFCS level, probably as a result of the way the classification was developed.

The high number of devices contrasts with the level of evidence reported in the literature. Gait orthoses have been shown to have a positive effect on gait but the long-term effects are unknown . Ankle-foot orthoses prevent equinus (low level evidence) but have not been shown to reverse deformities . There is little evidence on the effectiveness of corsets, hip and upper limb orthoses .

1.4.3

Paramedical input

The results of this study showed that the children in the sample had a median of 3.85 sessions of rehabilitation (all types) per week, ranging from two sessions for level I children to five sessions for level V children. Almost all the children had physiotherapy, half had occupational therapy and one third had speech therapy and psychomotor rehabilitation. Surprisingly, children with GMFCS level I (able to walk independently, where they want, including uneven surfaces, slopes and in crowds, able to run and jump despite slight limitations in speed, balance and coordination) had a lot of therapy: two sessions per week, 92.6% had physiotherapy, 35.2% had occupational therapy and 25.9% had speech therapy.

This high level of rehabilitation is in contrast with the levels of evidence in the literature. There is a moderate level of evidence for lower limb strengthening exercises and constraint induced therapy for the upper limbs . Other physiotherapy and occupational therapy techniques have not been shown to be effective for CP . Particularly, long-term therapy has not been shown to be justified. Neither speech therapy nor vision rehabilitation have been shown to be effective in this population, mostly because of the poor methodological quality of the studies. To our knowledge, no studies have evaluated the effectiveness of psychomotor rehabilitation in this population. About one third of the children were followed by a special educator and a psychologist but there are very few studies on the effectiveness of these disciplines.

1.4.4

Towards profiles of medical and paramedical input

The average profile of medical and paramedical input for a Breton child with CP can be established from our results: he or she receives on average almost four therapy sessions per week, including two sessions of physiotherapy and 0.5 of occupational therapy, has one chance in three of taking oral medication, one chance in three of receiving botulinum toxin injections within the year, 85% chance of having orthotics or adapted seating, almost one chance in two of having a mobility aid and consults a doctor (any type) an average of five times per year.

Since the quantity and frequency of therapies are mostly correlated with the GMFCS level, it would be possible to establish a management profile for each GMFCS level. However, the small number of subjects in each level makes it difficult to establish precise profiles but there are tendencies. Level I children have at least two rehabilitation sessions per week, 25% take medication, 78% use an orthotic device and 2.7% use a gait aid. Stage II children have on average four rehabilitation sessions per week, 46% take medication, 78% use an orthotic device and 37% have a mobility aid (mostly a manual wheelchair). The profiles of stage III and IV children are quite similar, with 3.5 and five rehabilitation sessions per week, 50% take medication, almost all have mobility aids (94% and 100%) and they use respectively an average of two and 3.5 orthotic devices. They receive more botulinum toxin injections than the other levels (50% and 44% respectively). Stage V children have an average of five rehabilitation sessions per week, 50% take medication but only 17% receive regular botulinum toxin injections, and they have 3.5 orthotic devices. They have more input from special educators than the other levels (56%). Because of the low correlation between GMFCS level and medical follow-up, this does not seem to be a particular characteristic of the general management.

The clinical pertinence of these results is reinforced by the prognostic capacity of the GMFCS. Indeed, this scale has been shown to be very stable over time with few children changing level from the age of 12 years . By including more children and by comparing them to an adult population, these data could be used to predict changes in management within a GMFCS level. This would help professionals to prepare families with regard to potential changes in the management of their child.

1.4.5

Study limitations

One of the first limitations of the study is the questionnaire addressed to the parents which is a source of possible errors. This method was chosen, rather than a questionnaire addressed to the professionals who often lack time, in order to increase the response rate. However, the good coherence between the devices described by the parents of the children with CP and the definitions of the GMFCS levels indicates a good quality of parent replies. Another limitation is the small sample size making an analysis of the five classes or as a function of age difficult, particularly since the groups for levels III, IV and V of the GMFCS consisted of only 16 and 18 children. Although our population is similar to that of other studies, we cannot exclude a selection bias related to difficulties in accessing the Medico-Educational Institutions, only 5% of children in the sample were registered with them. Certain results such as the low level of medication should therefore be interpreted with caution. Following this observation, one of the aims of this observational study is to better reach organizations such as the Institute for Motor Education and the Medico-Education Institute, in order to have a sample which is more representative of the population. Lastly, although the management strategies were reported via a network of many doctors, these strategies reflect practice in a region of France and therefore the results cannot be generalized to the whole country.

1.4.6

Impact of reported management strategies

This study reports a large gap between the frequency of rehabilitation and equipment provision and the level of evidence for these treatments. In contrast, botulinum toxin injections have been clearly shown to be effective on spasticity but only one third of children receive them regularly. This contrast between evidence and actual practice likely has several explanations: firstly, poor methodological quality of studies as reported in numerous literature reviews. Many treatments are available and used, as confirmed in this study, but their effects are currently unknown. Secondly, methodological and material difficulties in designing projects to evaluate the effectiveness of these treatments on the functions they aim to restore and their long-term effects. Thirdly, there is a gap between studies carried out on population averages and daily practice with a particular child or family. It is, however, difficult to justify an “intensive” and long-term management strategy as reported in this study without evidence other than the experience and good will of the professionals involved. Equally, in the current climate of reorganization of the reimbursement of therapy in France, it is difficult to justify the cost of medical and paramedical input for these children when there is no evidence of their effectiveness . The efforts which have been made internationally over the past 20 years to demonstrate the effectiveness of therapies in this population must be continued in order to highlight ‘optimal’ practices, i.e. those which are most likely to make a difference while at the same time respecting the child’s daily life and that of his or her family.

2.2.1

Type d’étude

Il s’agissait d’une étude transversale descriptive par autoquestionnaire.

2.2.2

Population d’étude

Entre février 2010 et juin 2011, 450 questionnaires avec enveloppes prépayées ont été envoyés à 40 médecins des quatre départements bretons assurant le suivi des enfants paralysés cérébraux en consultation (neuropédiatres, médecins de MPR, chirurgiens pédiatres, médecins des instituts médico-éducatifs, instituts d’éducation motrice, centres d’aide médicosocial précoce, services d’éducation et de soins spécialisés à domicile). Le questionnaire a été remis aux parents d’enfants paralysés cérébraux par l’intermédiaire de ce réseau. Un travail régulier de relance téléphonique et électronique auprès des médecins a été entrepris pendant la même période.

2.2.3

Le questionnaire

L’objectif de ce questionnaire était de définir les soins actuels des enfants paralysés cérébraux, réalisant une photographie, à un instant donné de leurs soins. Les questions posées aux parents portaient sur l’ensemble des prises en charge de l’enfant, au moment où le questionnaire était rempli : traitements per os (report de l’ordonnance sur le questionnaire), injections de toxine botulique (dans les six à 12 mois et localisation du (des) membre(s) injecté(s)), pompe à Liorésal, appareillage de déambulation et de posture, suivi médical et paramédical (fréquence du suivi). Le questionnaire suivait les recommandations de McColl et al. et répondait aux critères suivants :

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  chaque questionnaire a été accompagné d’une lettre d’explication, adressée aux parents, leur expliquant les objectifs de ce questionnaire, leur garantissant liberté de réponse, confidentialité et anonymat ;

  
  
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  il était suffisamment court pour pouvoir être renseigné en moins de 15 minutes et un niveau de langage simple était utilisé. Les termes techniques ont été expliqués par des photos (exemple : déambulateur, coquille de station assise…) ;

  
  
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  la présentation a été effectuée de façon à être la plus aérée possible, au moyen de tableaux, avec un maximum de cases à cocher. Pour que les parents puissent s’exprimer davantage s’ils le désiraient, un champ libre a été placé en fin de questionnaire ;

  
  
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  une première page a été effectuée à l’attention des médecins, afin de confirmer le diagnostic de PC (seul critère d’inclusion dans l’étude), le type de PC (spastique unilatérale, spastique bilatérale, dyskinétique, ataxique ou autre), les membres atteints, le niveau cognitif (pas de retard mental, retard mental léger ou sévère). Dans l’objectif de regrouper cette première page et la suite du questionnaire, un numéro d’anonymisation a été attribué à chaque questionnaire.

2.2.4

La classification GMFCS

Il était demandé aux parents de classer leurs enfants selon la GMFCS. Pour les tranches d’âge 4–6 et 6–18 ans, deux textes différents de GMFCS simplifiés ont été utilisés. La fiabilité de cette procédure a déjà été démontrée pour cette tranche d’âge . Ces deux textes ont été traduits par un kinésithérapeute anglais natif. Pour les enfants de deux à quatre ans, le texte utilisé était celui de la version complète de la GMFCS précédemment traduite en français . Dans le questionnaire remis aux parents, le stade V de la classification, correspondant aux enfants dont la fonction motrice est la plus limitée, a été placé en haut des propositions, afin que les parents n’aient pas à lire la liste des activités que leur enfant ne peut effectuer, avant d’atteindre la description de son niveau fonctionnel. Les stades suivants étaient explicités par ordre décroissant.

2.2.5

Analyse statistique

La saisie des données a été effectuée avec le logiciel EPIDATA ^® et l’analyse statistique avec le logiciel SAS 9.1 ^® . Les différentes caractéristiques de l’échantillon d’enfant PC ont été décrites en fonction de leur prise en charge médicale et paramédicale. Une analyse univariée a été réalisée en testant s’il y avait une tendance à l’augmentation ou à la diminution de la prise en charge considérée en fonction du stade GMFCS. Les variables d’intérêts étaient soit des variables qualitatives (prise en charge par un kinésithérapeute [oui/non]…), soit des variables quantitatives (nombre de consultations, nombre d’appareillages…). Pour étudier la tendance entre les variables de soins qualitatives et le niveau GMFCS, un test de Cochran Armitage a été réalisé. Pour étudier la relation entre le nombre de soins (quantitatif) et le niveau GMFCS, un test de corrélation de Spearman a été effectué. Un degré de significativité de 0,05 a été retenu comme significatif.

2.3.1

Population

Sur 450 questionnaires envoyés aux médecins, 133 ont été reçus, soit un taux de réponse de 29,6 %. Le Tableau 1 présente les caractéristiques des répondants. Les personnes ayant répondu au questionnaire étaient le plus souvent la mère (82,7 %). Le sex-ratio était de 1,8 en faveur des garçons et l’âge moyen était de 8,4 ans. 33,8 % des enfants avaient entre deux et six ans, 34,6 % entre six et 12 ans et 29,3 % entre 12 et 18 ans. Trente pour cent des enfants avaient une PC de type spastique unilatérale, 51,7 % de type spastique bilatérale, 17,5 % de type dyskinétique et 0,8 % de type ataxique. 40,6 % des enfants avaient un niveau fonctionnel correspondant au stade I de GMFCS, 20,3 % au stade II, 12,03 % au stade III et 13,53 % aux stades IV et V. 28,5 % des enfants présentaient un retard mental déclaré par le médecin (21 % de retard mental léger, 7,5 % de retard mental sévère).

Tableau 1

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