Abstract
Objective
Evaluate from the literature, the evidence of comparative efficiency of non-surgical treatments (orthotics or head repositioning therapy) in posterior positional plagiocephaly.
Material and methods
Systematic review from scientific articles (original cohort studies and review of literature), published in French or in English, searched on five online literature data bases, comparing non-chirurgical treatments (repositioning and orthotics therapy) for deformational plagiocephaly. A standardized method guidelines (Critical Review Form–Quantitative Studies) has been used.
Results
Only 11 cohort studies met the inclusion criteria and six reviews of literature were analyzed. Many biases have been identified, most of the time, favoring the repositioning groups (older infants and plagiocephaly more severe).
Conclusions
Several different orthotics seem to correct head deformities better and faster than repositioning protocols. Evaluation methods, treatment indications and long-term efficacy should be clarified. Studies about treatment risks are warranted.
Résumé
Objectif
Évaluer, à partir des données de la littérature, les preuves de l’efficacité comparée des techniques non chirurgicales de prise en charge de la plagiocéphalie postérieure positionnelle (techniques orthétiques ou protocoles de repositionnement).
Méthode
Une revue de littérature a été réalisée à partir d’articles (revues de littérature ou études de cohortes) publiés en anglais ou en français, recensés sur cinq bases de données sur la comparaison des protocoles non chirurgicaux de traitement de la plagiocéphalie postérieure positionnelle. Une grille de lecture standardisée a été utilisée (Critical Review Form–Quantitative Studies).
Résultats
Six revues de littérature ont été retrouvées sur le sujet ainsi que 12 articles originaux parmi lesquels 11 ont été retenus. De nombreux biais ont pu être mis en évidence, le plus souvent en faveur du repositionnement (les enfants étaient souvent plus âgés et avec une plagiocéphalie plus sévère dans les groupes appareillés).
Conclusion
Plusieurs types d’orthèses crâniennes semblaient aboutir à des corrections plus importantes et plus rapides que les protocoles de repositionnements. Une clarification des méthodes d’évaluation et des indications de traitement ainsi que des évaluations des risques et du maintien de l’efficacité à plus long terme seront nécessaires.
1
English version
1.1
Introduction
The term “plagiocephaly” stems from the greek “plagios” which means “oblique” and from “kephalê” meaning “head”. Positional plagiocephaly leads to cranial deformity, typically resulting in a parallelogram-shaped skull, with occipital flattening on one side, anterior shifting of the homolateral ear and prominence of the forehead, or even of the homolateral zygomatic region . It contrasts with the symptomatic plagiocephalies of craniostenosis by premature unilateral closure of lambdoid or coronal suture.
Differential diagnosis is clinical through the skull form and the deviations of the homolateral cheekbone and ear. In case of doubt, X-rays or even a scanner with bone window setting are performed to confirm the cranial suture opening .
Posterior positional plagiocephaly develops progressively during the first weeks of life owing to supine position whereas the cervical tonus does not allow the infants changing their position . It can also be the consequence of external intrauterine pressures.
The incidence of posterior positional plagiocephaly has dramatically increased some 20 years ago following the new recommendations of the American Academy of Pediatrics in 1992 prohibiting the infants’ prone position during sleep. Indeed, at the same time as sudden infant death syndrome decreased for more than 40% (from an incidence of 1.2/1000 births in 1992 to 0.56/1000 in 2001 ), the number of young children presenting with posterior plagiocephaly grew from one birth out of 300 to one out of 60 .
The most frequently quoted consequences of posterior positional plagiocephaly are of esthetic order . Although less frequent, other more severe consequences may appear. They may affect the relationships between the infants and their parents, and later the psychological status of the children . No rigorous study investigates the further development of children presenting with posterior positional plagiocephaly , but a few authors reveal a more important rate of development delay and of the need to perform schooling adaptations in children with plagiocephaly. On the other hand, the frequency of facial and maxillofacial deformity, cervical scoliosis, visual and hearing difficulties seems more important in children who presented plagiocephaly .
There is no “gold standard” for evaluating and quantifying the importance of plagiocephaly. The methods of evaluation may be based on subjective scales worked out by the authors , based on purely visual evaluations, including sometimes the parents’ opinion . There are also some objective evaluations based on anthropomorphic measurements . Their advantage is to be more easily reproducible than the three-dimensional pictures (3-D) and to prevent the infant from exposure to the radiations used during X-rays or scanning, but they lead to a loss of information because they are two-dimensional , and their reproducibility may be tricky in some cases .
Several means are considered to allow 3-D evaluation. Hutchison et al. are using a 3-D picture system in association with anthropomorphic measurements. Mottolese et al. opt for a 3-D reconstruction starting from a cerebral tomodensitometry (TDMc). As regards surface scanner, it was the subject of several studies with the production of a mathematical and statistical method of results comparison in order to assess the efficacy of the management .
The treatment options are conditioned by the selected evaluation method and remain dependent on the examiner. Besides, according to Lee et al. , these discrepancies are important between the neurosurgeons and the plastic surgeons. To evaluate the management efficacy, these same measures are being used, as well as the degree of satisfaction of the parents or of the examiner . Some authors, like Losee et al. assess it upon the changes of the head position during sleep (which would be secondary to an improvement of posterior plagiocephaly).
The non-surgical management techniques of posterior positional plagiocephaly are the repositioning programs, cranial orthosis (with or without associated physical therapy) and sometimes a wait-and-see policy with the hope of spontaneous correction. If the decision of any management is taken, it must be made early in order to be supported by the remodeling capacities due to the growth of the skull (85% during the first year of life) .
Repositioning consists in alternating the positions of the head during bedding, while limiting the predisposing factors (in particular the time spent with a posterior resting surface) and increasing the time spent on the tummy when not sleeping (“tummy time”).
The first descriptions of cranial orthosis of helmet type go back to the end of the 1970s with the works by Clarren et al. . Despite a lack of methodology quality , most of the studies are in favor of a good short-term efficacy (helmet or other ), as is shown in the work aiming the certification of the helmets by the American Food and Drug Administration (FDA) , in particular for the aged children presenting with a severe plagiocephaly, beyond 6 months’ delay and if the response to the conservative treatment is insufficient . However, a retrospective study suggests that there would be no long-term efficacy of the helmet , and the recent study by Flannery et al. reminds that the helmet efficacy would need new studies, of high standard of evidence.
The helmets are made from a molding on a semi-rigid material; most of them have an expansion zone facing the occipital flattening and a head-rest at level of the forehead bulge. Teichgraeber et al. propose a cranial orthosis made of bands (DOC bands) which would allow reducing significantly ( P < 0.001) the asymmetry of the base of the skull.
The complications secondary to the use of cranial orthosis are rare . They may lead to contact dermatitis, cutaneous irritation, pressure sore, cervical trauma due to the displacement of the gravity centre, or psychological consequences at the children, and especially at the parents . In the Anglo-Saxon countries, a controversy exists concerning the cost/efficacy relationship of cranial orthosis, which was reinforced by the non-reimbursement of the helmets (graded Class II by the FDA) and the related financial impact . In France, their cost is of about 600 € for the first, then 350 € for the second .
In this context, we decided to perform a literature review of the articles comparing the efficacy of the non-invasive management techniques of posterior positional plagiocephaly.
The objective was to provide some elements of answer in order to learn more about the most efficient non-surgical management of posterior positional plagiocephaly.
1.2
Methods
To perform this literature review, a protocol was designed to determine the objectives and the search strategy (selection of the data bases and of inclusion and exclusion criteria). A validated interpretation form, Critical Review Form ( Appendix 1 ), worked out for the analysis of quantitative studies , was used to evaluate in a systematic and reproducible way the relevance of the selected articles.
1.2.1
Search strategy
At first, literature review on the subject was searched in the Cochrane Library, and then in the following data bases: MEDLINE (PubMed), Springerlink, ScienceDirect, Journals@ovid, and Google Scholar.
The Medical Subject Headings (MeSH) was used for the keywords definition, allowing a truncation which prevented from any risk of article omission. For the data base search we used the following:
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in French: plagiocéphalie ET orthèse, or plagiocéphalie ET casque;
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in English: plagiocephaly AND ortho* (for orthotics or orthosis) or plagiocephaly AND helmet.
1.2.2
Criteria for the selection of articles
The selected articles had to:
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be written in English or in French;
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be published after 1992 (year of the American Academy of Pediatrics guidelines publication);
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be literature reviews or original quantitative studies;
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concern children of less than 18 months of age;
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treat posterior positional plagiocephaly.
The articles were excluded if:
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children with craniostenosis had been included;
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it was an isolated Abstract;
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it was a case study or an expert’s opinion (not based on a clinical quantitative study).
1.2.3
Evaluation method
All selected articles were analyzed with the help of a validated interpretation form ( Appendix 1 ) for which a user’s guide was worked out by the authors.
1.3
Results
1.3.1
Studies selection
This search allowed referencing 18 different articles meeting the inclusion criteria and treating the efficacy comparison of two non-surgical management methods of posterior positional plagiocephaly. Among these, there were six literature reviews . Except for the review by Xia et al. , none of these included only comparative articles. The review by Xia et al. evaluates exclusively the comparison between the repositioning techniques and the use of cranial orthosis of helmet type.
We were able to collect 12 original articles ( Table 1 ) dealing with the comparison of the management techniques of posterior positional plagiocephaly.
| Authors | Design | Patients | Intervention | Analysis | ||||
|---|---|---|---|---|---|---|---|---|
| n | Mean age at treatment start | Intervention | Treatment duration | Measurements | Contamination / co-intervention | A and B comparable | ||
| Graham et al., 2005 | Case/control Retrospective Non-randomised | n = 298: A: 176 B: 159 | A: 4.8 months B: 6.6 months | A: repositioning B: helmet Choice by the parents | A: 3.5 months B: 4.2 months ( P = 0.024) | Objective | Yes / possible | No, B second intention |
| Hutchison et al., 2000 | Cohort Prospective Randomised | n = 126: A: 61 B: 65 | A and B comparable | A: repositioning + stretching B: idem + Safe T Sleep Physical therapy if stiff neck associated | A = B: 12 months | Objective But non-validated scale | No/yes (+ physical therapy if stiff neck) | Yes, randomised |
| Lipira et al., 2010 | Cohort Prospective Non-randomised | n = 70: A: 35 B: 35 | A: 4.8 months B: 4.9 months Comparable | A: repositioning B: helmet Choice by the parents | A: 5.2 months B: 3.1 months | Objective | No (excluded)/possible | Yes, matched groups |
| Losee et al., 2007 | Case/control Retrospective Non-randomised | n = 105: A: 100 B: 45 | A: 6.5 months B: 7.6 months (2nd intention) | A: repositioning B: helmet | A: ? B: 3.7 months | Subjective | Systematic in A/ ? | No, B 2nd intention |
| Loveday and de Chalain, 2001 | Cohort Prospective Randomised | n = 74: A: 45 B: 29 | A: 8.8 months B: 8.5 months Comparable | A: repositioning B: helmet | A: 63.7 weeks B: 21.9 weeks | Objective | Risk/? | No, severity B > A |
| Moss, 1997 | Cohort Prospective Non-randomised Historical controls | n = 112: A: 66 B: 46 | A: 6.4 months B: 5.9 months Comparable | A: repositioning + stretching B: cranial bands | A: 4.5 months B: ? | Objective but different for the 2 groups | No/? | No, historical group |
| Mulliken et al., 1999 | Cohort Prospective Non-randomised | n = 114: A: 63 B: 51 | A: 5.6 months B: 5.4 months Comparable | A: repositioning with foam blocks B: helmet Choice by the parents | A: 4.8 months B: 4.6 months | Objective But only in 17 in A and 36 in B | No/? | No, different size and many lost to follow-up |
| Plank et al., 2006 | Cohort Prospective Non-randomised | n = 224: A: 17 B: 207 | ? But between 3 and 12 months | A: repositioning (if orthosis refused) B: cranial bands | A = B: 4 months approximately | Objective | Risk/? | Yes, but sizes very different |
| Pollack et al., 1997 | Cohort Prospective Non-randomised | n = 69: A: 69 B: 34 | ? helmet 2 to 3 months later | A: repositioning ± orthosis B: helmet | A: 2-3 months B: ? (until symmetry) | Subjective | Systematic in B/ possible | No, B 2nd intention |
| Rogers et al., 2008 | Cohort Prospective Non-randomised Historical controls | n = 47: A: 23 B: 24 | A: 88 days B: 96 days Comparable | A: repositioning + stretching ± orthosis B: cranial cup | A: 61.6 days B: 56.3 days | Objective | No/yes in A | Yes, matched |
| Teichgraeber et al., 2004 | Cohort Prospective Non-randomised | n = 380: A: 132 B: 248 | ? | A: repositioning B: helmet | Non-defined | Objective | ?/? | No, because plagiocephaly and brachycephaly in A |
| Vles et al., 2000 | Cohort Prospective Non-randomised | n = 105: A: 39 B: 66 | ? | A: repositioning B: helmet | A: 5.6 months B: 1.2 months | Subjective | ?/? | No, severity B > A |
1.3.2
Evidence level of the original articles
The randomized and controlled studies are considered as the methodological gold standard of the studies evaluating the efficacy of therapeutic management. Unfortunately, this search did not allow finding any article of this type on the subject.
Among the 12 selected articles ( Table 1 ), 10 articles were based on prospective studies of cohorts , two of them being based on retrospective studies .
1.3.3
Methodological quality of the original articles
1.3.3.1
Subject definition
All articles selected for this literature review deal with the comparison of the non-surgical methods of management of posterior positional plagiocephaly. They compare a repositioning method possibly associated with stretching exercises of the cervical muscles to a method using orthosis.
The objective of each study is clearly defined.
1.3.3.2
Sample and recruitment
The number of children included in the different groups of the 12 studied articles is extremely variable ( Table 1 ). The greatest cohort of children with cranial orthosis counts 248 children , the smallest include 24 children .
The sample size is justified in none of the studies.
The modalities of distribution among the groups are fickle ( Table 1 ). In two studies the children are randomized , in five others the distribution among the groups is decided by the parents, sometimes upon proposal of the examiners (if age less than 4 months, proposal of repositioning; if age greater than 6 months, a helmet is suggested; between both proposals, free choice of the parents). Historical control groups are used in two studies , two protocols propose a repositioning program to all included children and use cranial orthosis on second intention in case of failure of this program. In Graham et al., the children without any improvement of their plagiocephaly following 2 to 3 months of repositioning switch to the orthosis group. Moreover, in this same study , mean age as well as age at start and at end of treatment are different according to the groups.
The inclusion criteria in the different groups are not systematically detailed .
The presence of stiff neck is highlighted by some authors with a frequency varying from 14.5% , to 100% of the included children and leads then to a specific physical therapy management. In the article by Losee et al. , neck stiffness is described as a risk factor of plagiocephaly and of non-response to repositioning treatment.
Some authors specify that parents gave their consent .
Only two articles specify a priori the severity of the included plagiocephalies. The study by Plank et al. concerns the moderate to severe deformities, whereas that by Moss is limited to little severe plagiocephalies. Otherwise, in three studies , the severity of the plagiocephaly in the children of the orthosis group at study start is more important than in those of the repositioning group.
1.3.3.3
Variables measured
The variables are measured either by visual subjective means by the parents and/or by the examiner , or by objective means ( Table 1 ). In this purpose, two-dimensional anthropomorphic measurements are used , pictures being systematically taken for some of them with 3-D reconstruction . In other cases, 3-D evaluations are performed with the help of a surface scanner .
Some authors use non-validated subjective scales based on anthropomorphic – or only visual – evaluations to grade the plagiocephaly severity. They include four, nine, and eleven levels.
In the article by Losee et al. , the final evaluation is based on the ability to change position during sleep, which would be secondary to a correction of the plagiocephaly.
It is worth noting that in the study by Mulliken et al. , the anthropomorphic measurements are not performed on the whole cohort. Indeed the measurements concern 76 children out of 114 included during initial evaluation, then only 53 during final evaluation (36 of the 51 children included in the orthosis group, and 17 of the 63 children of the repositioning group). The number of lost to follow-up is very important.
On the other hand, the anthropomorphic measurements realized by Moss are not equivalent to those realized a few years earlier in the control group (historical) treated by cranial orthosis.
1.3.3.4
Intervention
The modalities of intervention are very different according to the selected studies ( Table 1 ). Most of the studies compare a repositioning program to a treatment by cranial orthosis of helmet type made of thermally malleable material or made from cranial bands . But Rogers et al. assess a different cranial orthosis called “cranial cup”. It is presented like a concave, custom-made foam pillow, adjusted as the skull grows, possibly with a high dorsal resting surface in order to limit the difference of level between the shoulders and the neck, and hence to reduce the cervical bending. Hutchinson et al. describe a system of positioning on the mattress (the Safe T Sleep).
In some articles, the detail of the repositioning protocols is missing .
The daily wear time of the helmets is defined (between 20 and 23 hours a day) , except for the cranial orthosis of Rogers et al. .
The duration of intervention is not defined in all studies ( Table 1 ), it varies from 1.2 months (i.e. 5.4 weeks) to 63.7 weeks . The treatment duration in the repositioning and orthosis groups is identical only in two protocols . In the other cases, it is shorter in the group treated by orthosis . For example, in the work by Loveday and de Chalain , it is clearly higher to that of the orthosis group (63.7 vs. 21.3 weeks). The duration of intervention is sometimes decided by the parents themselves .
Age lower than 1-year-old is an inclusion criteria in all studies except in those of Losee et al. (< 18 months), Mulliken et al. (< 10 months), and Rogers et al. (< 4 months). The treatment by repositioning or orthosis starts at similar age in six studies . In the protocols using orthosis on second intention , the age at repositioning treatment start is significantly lower (4.8 months vs. 6.6 and 6.5 months vs. 7.6 for example).
Some authors propose physical therapy management to all included children or a program of stretching . Others appeal to these programs only for the repositioning group . Finally, two articles mention that no physical therapy management is proposed.
1.3.3.5
Results presentation
The statistical validity of the studied articles is satisfying in most of them , but statistics are missing or little detailed in others.
Four studies count the withdrawals from the inclusion group.
1.3.4
Efficacy of the conservative treatment
We have excluded from our analysis the study by Teichgraeber et al. as the studied population included also children presenting with brachycephaly. On the other hand, the article does not provide the detail of the comparison between the sub-groups treated by orthosis or exclusively by repositioning in the children presenting with plagiocephaly.
The results of the 11 selected studies are summarized in Table 2 . Their comparison must be considered with caution since the methodology varies from one study to another. Among these 11 studies, three report comparable results between the repositioning group and the orthosis group. The others report a better efficacy of the orthotic management.
| Authors | Group A | Group B | Treatment duration | Results Group A | Results Group B | P | Conclusion |
|---|---|---|---|---|---|---|---|
| n | n | ||||||
| Graham et al., 2005 | 176 | 159 | A: 3.5 months B: 4.2 months ( P = 0.024) | Efficacy in 139 children, no efficicacy in 37. Reduction of DD = 0.55 cm | Reduction of DD = 0.71 cm | P < 0.001 | Repositioning efficient if PPP mild. If PPP moderate to severe, helmet more efficient |
| Hutchison et al., 2000 | 61 | 65 | A = B: 12 months | CI = 89.5 and DD = 105.6 | CI = 88.4 and DD = 105.7 | P CI = 0.32 and P DD = 0.76 | No difference. STS does not improve the efficacy of repositioning |
| Lipira et al., 2010 | 35 | 35 | A: 5.2 months B: 3.1 months | Reduction of asymmetry = 0.5% | Reduction of asymmetry = 0.9% | P = 0.02 | More important improvement with shorter treatment duration in the helmet group |
| Losee et al., 2007 | 100 | 45 | B: 3.7 months | Reduction of subjective asymmetry = 1.31% | Reduction of subjective asymmetry = 3.11% | P < 0.05 | Helmet more efficient than repositioning alone |
| Loveday and de Chalain, 2001 | 45 | 29 | A: 63.7 weeks B: 21.9 weeks | CVAI modification = 1.9 % | CVAI modification = 1.8 % | Efficacy but treatment duration Group A 3 times longer | |
| Moss, 1997 | 66 | 46 | A: 4.5 months | Improvement in 65/66 children: mean CVAI decreases from 9.2 mm to 4.7 mm | Historical group | Not comparable | Comparable results in the 2 groups |
| Mulliken et al., 1999 | 63 | 51 | A: 4.8 months B: 4.6 months | DD = 1 cm | DD = 0.6 cm | P < 0.001 | Efficacy in both groups but improvement statistically more important and quickly than orthosis |
| Plank et al., 2006 | 17 | 207 | A = B: 4 months approx. | Worsening in 30% of patients | Better symmetry in all measurements in 96.3% of patients | Helmet more efficient than repositioning alone | |
| Pollack et al., 1997 | 69 | 34 | A: 2–3 months B: not defined (until symmetry) | Excellent results in all except in 5 | |||
| Rogers et al., 2008 | 23 | 24 | A: 61.6 days B: 56.3 days | DD from 9.0 to 8.0 mm | DD from 11.2 to 3.5 mm | P < 0.001 | Cranial cup significantly more efficient than repositioning |
| Vles et al., 2000 | 39 | 66 | A: 5.6 months B: 1.2 months | P < 0.01 | |||
Hutchison et al. conclude that the system of positioning on the Safe T Sleep (STS) mattress associated with a repositioning program does not improve the cranial symmetry more than the repositioning program alone. Whatever the group, 80% of the included children benefit from an important improvement. The study by Moss highlights an improvement of the cranial asymmetry in the group of infants benefiting from physical therapy and repositioning comparable to the results of a group of children treated by cranial bands several years earlier. Loveday and de Chalain obtain anthropomorphic measurements comparable between the repositioning group and the helmet group (modification of the asymmetry index of 1.9% and 1.8%, respectively).
Despite the protocols heterogeneousness, the results seem to be in favor of the orthotic treatment. The authors of eight studies describe a more important efficacy of the orthotic management, significant ( P < 0.001) in the studies by Graham et al. , Mulliken et al. , and Rogers et al. for instance. These results concern orthosis of helmet type to be worn by day and by night or the orthosis of concave pillow type developed by Rogers et al. .
Early management (before the age of 1 year) allows a better correction of the cranial deformity as shown by Hutchison et al. and Pollack et al. , but also in three literature reviews .
Among the six literature reviews analyzed, only that of Singh and Wacogne does not demonstrate a formal evidence of the benefit of the helmet compared to repositioning or to absence of treatment when posterior positional plagiocephaly is moderate. The other authors find solid arguments for the efficacy of the management by helmet. Orthosis would probably have a greater and quicker efficacy than repositioning for Robinson and Proctor and for Mc Garry et al. . The articles by Robinson and Proctor and by Lima make a clarification on the literature data concerning posterior positional plagiocephaly and its non-surgical management. They bring to light the importance of prevention and parental education, and in conclusion they propose the use of cranial orthosis if the repositioning protocols are not sufficient to correct plagiocephaly.
Xia et al. precise that there is a general consensus for the implementation of repositioning protocols if the severity of the plagiocephaly is not too important, without it being supported by solid bibliographical references. The works by Singh and Wacogne , Bialocerkowski et al. and Mc Garry et al. focus on the efficacy of cranial orthosis. The latter work has the particularity to be interested in the objective means of plagiocephaly evaluation. The review by Bialocerkowski et al. concludes that repositioning and physical therapy may reduce cranial asymmetry.
1.4
Discussion
The benefits of the orthosis have been under-estimated by several biases in some studies . Yet there seems to be a trend in favor of a greater efficacy of the correction of asymmetry by cranial orthosis (helmet, or custom-made pillow system) than by the repositioning programs. This was particularly clear in case of severe posterior positional plagiocephaly where the orthosis could correct better and faster. However, the studies present numerous limits.
1.4.1
Methodological limits
1.4.1.1
On the power of the studies
The selected studies present great disparities and a weak power of methodology. Among the six reviews indeed and the 12 retrieved original articles , only two trials were randomized.
1.4.1.2
On the evaluation of plagiocephaly
The evaluation of posterior positional plagiocephaly was also source of bias because of the absence of standardized criteria for the evaluation of the cranial asymmetry (no solid evidence for the validity and the reproducibility of the anthropomorphic measurements, insufficient evaluation of the surface scanners and loss of data secondary to the two-dimensional evaluation), as pointed out by most literature reviews .
The choice of measurement variables was different according to the studies: cranial vault asymmetry index , difference of the diagonals , cranial index , parents’ advice , visual perception , 3-D surface scanner .
The use of non-validated subjective scales , sometimes based only on visual evaluation , was problematic as it clearly limited the objectivity of the studies, as was the case for evaluations performed by only one examiner .
1.4.1.3
On the groups compared
As it is underlined in some studies , the modalities of distribution into the groups led sometimes to biases, especially when it was decided by the parents , possibly on proposal of the examiners.
The populations of the repositioning and orthosis groups of several studies were not homogeneous ( Table 2 ):
- •
historical control groups were used in two studies . The two groups were sometimes of much different size ;
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in the study by Mulliken et al. , the very high number of lost to follow-up (46 out of 63 in the repositioning group and 15 out of 51 in the orthosis group) has probably modified the results;
- •
in two studies , there was a bias of systematic contamination for the children treated by orthosis as they benefited previously of the repositioning program with non-satisfying results. This bias was also present in some children in the study by Loveday and de Chalain , as well as in 37 patients of the Graham et al. study . In the other studies, the contamination was avoided by excluding the children who switched from one group to another;
- •
there was a risk of co-intervention through the action of physiotherapists in case of neck stiffness , or through the non-systematical use of means of posture in the repositioning group .
Several types of bias have favored the repositioning group. In three studies , the plagiocephalies resistant to the repositioning treatment were secondarily included in the orthosis group. This has delayed the age of orthosis treatment start (6.6 vs. 4.8 months) and (7.6 vs. 6.5) , and has probably increased the mean severity of the plagiocephalies in this group, as with Graham et al. , Vles et al. or Loveday and de Chalain . Moreover, in some studies , the treatment duration of the orthosis group was shorter, up to three times as in the Loveday and de Chalain study .
These methodological limits allow making hypotheses explaining the results of the three studies finding comparable results in the repositioning and orthosis groups:
- •
in Hutchison et al. , the results with STS may be explained by a less important application of the repositioning program by the parents of the STS group children, although they had received the same information as the repositioning group alone;
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in Moss , anthropomorphic measurements are used in both groups, but with different anatomical markers, making the conclusions tricky to interpret;
- •
the treatment duration of the repositioning group in Loveday and de Chalain is nearly three times longer than for the orthosis group (63.7 weeks vs. 21.9 weeks).
1.4.2
Nature of the intervention
The physical therapy interventions are very rarely detailed. There is no precision as to whether osteopathy techniques are associated. The same applies to the repositioning protocols, of very little precision. This leads to suppose that the interventions in physical therapy and in the repositioning group are quite disparate among the articles, but also within a same study.
1.4.3
Financial issue
The cost of the cranial orthosis and their non-reimbursement by the Social Security systems (in France, United Kingdom and the United States) represent a problem, and this contributes to maintain the controversy on the orthosis indications .
1.4.4
Risks related to fittings
There are few details in the different articles on the risks related to fittings. The orthosis of pillow type or of positioning on the mattress reduce de facto the free mobility of the cephalic extremity of the infants. We must therefore wonder about the consequences of the restriction of these spontaneous movements. This restriction seems to reduce the cortical excitability and so the periods when the infant wakens during sleep which could have a facilitating role in the onset of the sudden infant death . In this case, the risk–benefit balance of the posterior positional plagiocephaly treatment should be re-evaluated, and the medico-legal aspect of this type of fitting should be taken into account.
This type of questioning does not apply to the helmet-type orthosis.
1.4.5
Conflict of interest
Several literature reviews question some authors as to whether some conflicts of interest may exist, as they would have links with some orthopedics companies, which could induce additional biases.
1.4.6
Indications and treatment efficacy
The natural evolution of posterior positional plagiocephaly is not perfectly known, although the evolution of the deformity seems to be spontaneously favorable after several years . Moreover, the evaluation methods are varied and there is no consensus about which one should be favored, or about the degree of plagiocephaly from which a treatment is necessary. We feel it essential to lay clear indications, based on an evaluation as much standardized as possible.
Among all selected studies, only two extend the follow-up beyond the end date of the plagiocephaly management programs. This does not allow asserting with certainty that the cranial asymmetry reduction obtained with the orthosis is maintained with time.
On the other hand, several studies recall a link between posterior positional plagiocephaly and cognitive and developmental delay ; no author studied the consequences of a reduction of the cranial deformities on these cognitive difficulties. If this was the case, the orthotic management would be legitimated even more in view of its fast action and correction efficiency.
1.4.7
Critical appreciation of our literature review
We met some difficulties all along our work, which impacted negatively its quality. A second reader applying the interpretation form to the quoted original articles and literature reviews would have improved the objectivity of the work. On the other hand, no cotation was associated to the evaluation of the selected articles what increased the risk of subjective appreciation.
We have only studied the posterior positional plagiocephalies in a concern of consistency. In spite of this, their mechanisms seem to be different according to a damage at the base of the skull or the presence of a stiff neck for instance. A better knowledge of these mechanisms could probably allow a more individual approach of the therapeutic responses to be proposed to these children.
1.5
Conclusion
Preventing posterior positional plagiocephaly seems essential to us. The results of the quoted studies need to be balanced by the biases inherent to the protocols used. However, and despite a few conflicting results, these articles seem to show a higher efficacy of the management by cranial orthosis of posterior positional plagiocephaly compared to repositioning, especially in moderate to severe plagiocephalies. The devices used during sleep time appear interesting as they limit the disadvantages related to the helmets and seem to be efficient, but further evaluations on their own risks and their efficiency would be needed.
On the other hand, the long-term maintenance of the correction of plagiocephaly following the end of management protocols is the main objective of these treatments, this being however not well known currently, as few studies investigate this subject.
Disclosure of interest
The authors declare that they have no conflicts of interest concerning this article.
2
Version française
2.1
Introduction
Le terme « plagiocéphalie » vient du grec « plagios » qui signifie oblique et de « kephalê » qui signifie tête. La plagiocéphalie positionnelle entraîne une déformation crânienne conduisant typiquement à un crâne en forme de parallélogramme, avec un méplat occipital d’un côté, un déplacement antérieur de l’oreille homolatérale et une proéminence du front, voire de la région zygomatique homolatérale . Elle s’oppose aux plagiocéphalies symptomatiques des craniosténoses par fermeture unilatérale prématurée d’une suture lambdoïde ou coronale.
Le diagnostic différentiel est clinique par la forme du crâne et les déviations de la pommette et de l’oreille homolatérales. En cas de doute, des radiographies, voire un scanner en fenêtres osseuses sont pratiqués afin de s’assurer de l’ouverture des sutures crâniennes .
La plagiocéphalie postérieure positionnelle se développe progressivement durant les premières semaines de vie à la faveur du décubitus dorsal alors que le tonus cervical ne permet pas aux nourrissons de changer de position . Elle peut aussi être la conséquence de contraintes externes appliquées in utero.
L’incidence de la plagiocéphalie postérieure positionnelle a augmenté de manière spectaculaire il y a une vingtaine d’années à la suite des nouvelles recommandations de 1992 de l’Académie Américaine de Pédiatrie qui prohibaient la position en décubitus ventral des nourrissons durant leur sommeil. En effet, de façon parallèle à la diminution du syndrome de la mort subite du nouveau-né de plus de 40 % (d’une incidence de 1,2/1000 naissances en 1992 à 0,56/1000 en 2001) , le nombre de jeunes enfants présentant une plagiocéphalie postérieure est passé d’une naissance sur 300 à une sur 60 .
Les conséquences de la plagiocéphalie postérieure positionnelle les plus souvent évoquées sont esthétiques . Bien que plus rares, d’autres conséquences plus graves peuvent apparaître. Elles peuvent retentir sur les relations entre les nourrissons et leurs parents puis sur l’état psychologique des enfants . Aucune étude rigoureuse n’étudie le développement ultérieur des enfants atteints de plagiocéphalie postérieure positionnelle mais certains auteurs mettent en évidence un taux plus important de retards de développement et de nécessité d’adaptations de la scolarité chez les enfants ayant présenté une plagiocéphalie. Par ailleurs, la fréquence de déformations faciales et maxillo-faciales, de scoliose cervicale, de difficultés visuelles et auditives semble plus importante chez les enfants ayant présenté une plagiocéphalie .
Il n’existe pas de « gold standard » pour l’évaluation et la quantification de l’importance de la plagiocéphalie. Les méthodes d’évaluation peuvent reposer sur des échelles subjectives élaborées par les auteurs , basées sur des évaluations purement visuelles, intégrant parfois l’avis des parents . Il existe aussi des évaluations objectives basées sur des mesures anthropométriques . Celles-ci ont pour avantages d’être plus facilement reproductibles que les photographies en trois dimensions (3D) et de ne pas exposer le nourrisson aux radiations utilisées lors des radiographies ou des scanners mais elles entrainent une perte d’informations car en deux dimensions et leur reproductibilité peut être délicate dans certains cas .
Plusieurs moyens sont envisagés pour permettre une évaluation en 3D. Hutchison et al. utilisent un système de photos en 3D associé aux mesures anthropométriques; Mottolese et al. optent pour une reconstruction en 3D à partir d’une tomodensitométrie cérébrale (TDMc). Quant au scanner de surface, il a fait l’objet de plusieurs travaux avec création d’un modèle mathématique et statistique de comparaison des résultats afin de juger de l’efficacité de la prise en charge .
Le choix des méthodes d’évaluation conditionne les options de traitement et reste dépendant de l’examinateur. D’ailleurs, selon Lee et al. ces divergences sont importantes entre les neurochirurgiens et les chirurgiens-plasticiens.
Pour évaluer l’efficacité de la prise en charge, ces mêmes mesures sont utilisées ainsi que le degré de satisfaction des parents ou de l’examinateur . Certains auteurs, comme Losee et al. l’évaluent sur les changements de position de tête pendant le sommeil (qui seraient secondaires à une amélioration de la plagiocéphalie postérieure).
Les prises en charges non chirurgicales de la plagiocéphalie postérieure positionnelle sont le repositionnement, les orthèses crâniennes (avec ou sans kinésithérapie associée) et parfois l’attentisme en espérant une correction spontanée. Si une prise en charge est décidée, elle doit être précoce afin de s’appuyer sur les capacités de remodelage liées à la croissance du crane (85 % au cours de la première année de vie) .
Le repositionnement consiste à alterner les positions de la tête lors du couchage, en limitant les facteurs favorisants (en particulier le temps passé avec un appui postérieur) et en augmentant le temps passé sur le ventre lors de périodes d’éveil ( tummy time ).
Les premières descriptions des orthèses crâniennes à type de casque remontent à la fin des années 1970 avec les travaux de Clarren et al. . Malgré un manque de qualité méthodologique , la plupart des études sont en faveur d’une bonne efficacité à court terme des orthèses crâniennes (casque ou autre ), comme le montre le travail en vue de la certification des casques par la Food and Drug Administration (FDA) américaine , en particulier pour les enfants âgés présentant une plagiocéphalie sévère, au-delà de six mois et si la réponse au traitement conservateur est insuffisante . Cependant, une étude rétrospective suggère qu’il n’y aurait pas d’efficacité du casque à long terme et la récente revue de Flannery et al. rappelle que l’efficacité du casque nécessiterait de nouvelles études de haut niveau de preuve.
Les casques sont réalisés sur moulage dans un matériau semi-rigide, la plupart ont une zone d’expansion en regard du méplat occipital et un appui au niveau du bombement frontal. Teichgraeber et al. proposent une orthèse crânienne élaborée à partir de bandes (DOC bands) qui permettrait de réduire significativement ( p < 0,001) les asymétries de la base du crâne.
Les complications secondaires à l’utilisation d’orthèses crâniennes sont rares . Elles peuvent entraîner des dermites de contact, une irritation cutanée, des escarres, des traumatismes cervicaux du fait du déplacement du centre de gravité ou des conséquences psychologiques chez les enfants et surtout les parents . Dans les pays anglo-saxons, il existe une polémique quant au rapport cout financier/efficacité des orthèses crâniennes. Celle-ci a été renforcée par la décision de non-remboursement des casques (classés en catégorie II par la FDA) et l’impact financier qui en découle . En France, le coût du casque est estimé à environ 600 € pour le premier puis 350 € pour le second .
Dans ce contexte, nous avons décidé de réaliser une revue de littérature des articles s’intéressant à la comparaison de l’efficacité des techniques de prises en charge non invasives de la plagiocéphalie postérieure positionnelle.
L’objectif était d’apporter des éléments de réponse afin de savoir quelle est la prise en charge non chirurgicale la plus efficace en cas de plagiocéphalie postérieure positionnelle.
2.2
Méthode
Pour réaliser cette revue de littérature, les objectifs et la stratégie de recherche (détermination des bases de données interrogées, des critères d’inclusion et d’exclusion) ont été définis dans un protocole. Une grille de lecture validée, Critical Review Form ( Annexe 1 ) élaborée pour l’analyse des études quantitatives a été utilisée afin d’évaluer de manière systématique et reproductible la pertinence des articles sélectionnés.
2.2.1
Stratégie de recherche
Dans un premier temps, des revues de littératures sur le sujet ont été recherchées dans la Cochrane Library. Puis les bases de données suivantes ont été interrogées : MEDLINE (PubMed), Springerlink, ScienceDirect, Journals@ovid et Google Scholar.
Le Medical Subject Headings (MeSH) a été utilisé pour la définition des mots clés, il a permis une troncature pour ne pas risquer d’omettre d’article. Nous avons utilisé pour la recherche dans les bases de données :
- •
en français : plagiocéphalie ET orthèse ou plagiocéphalie ET casque ;
- •
en anglais : plagiocephaly AND ortho* (pour orthotics ou orthosis) ou plagiocephaly AND helmet.
2.2.2
Critères de sélection des articles
Les articles sélectionnés devaient :
- •
avoir été rédigés en anglais ou en français ;
- •
avoir été publiés après 1992 (date des recommandations de l’Académie américaine de pédiatrie) ;
- •
être des revues de littérature ou des études quantitatives originales ;
- •
concerner des enfants de moins de 18 mois ;
- •
traiter de plagiocéphalie postérieure positionnelle.
Les articles étaient exclus si :
- •
des enfants présentant une craniosténose avaient été inclus ;
- •
il s’agissait d’un abstract isolé ;
- •
il s’agissait d’étude de cas ou un avis d’expert (ne reposant pas sur une étude clinique quantitative).
2.2.3
Méthode d’évaluation
Tous les articles sélectionnés ont été analysés à partir d’une grille de lecture ( Annexe 1 ) validée pour laquelle un guide d’utilisation a été élaboré par les auteurs.
2.3
Résultats
2.3.1
Sélection des études
Cette recherche a permis de référencer 18 articles différents remplissant les critères d’inclusion et traitant de la comparaison de l’efficacité de deux méthodes de prise en charge non chirurgicales de la plagiocéphalie postérieure positionnelle.
Parmi eux, il y avait six revues de la littérature . En dehors de la revue de Xia et al. , aucune n’inclut que des articles comparatifs. Celle-ci évalue uniquement la comparaison entre les techniques de repositionnement et l’utilisation des orthèses crâniennes à type de casque.
Nous avons pu collecter 12 articles originaux ( Tableau 1 ) s’intéressant à la comparaison des méthodes de prise en charge de la plagiocéphalie postérieure positionnelle.
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