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Positional Cranial Deformation In Children: A Plea For The Efficacy Of The Cranial Helmet In Children
Journal Pre-proof Positional Cranial Deformation In Children: A Plea For The Efficacy Of The Cranial Helmet In Children Thiebaud Picart MD Pierre-Aurelien Beuriat MD Alexandru Szathmari MD PhD Federico Di Rocco MD PhD Carmine Mottolese MD PhD
PII:
S0028-3770(20)30001-1
DOI:
https://doi.org/doi:10.1016/j.neuchi.2019.10.011
Reference:
NEUCHI 1050
To appear in:
Neurochirurgie
Please cite this article as: Picart T, Beuriat P-Aurelien, Szathmari A, Rocco FD, Mottolese C, Positional Cranial Deformation In Children: A Plea For The Efficacy Of The Cranial Helmet In Children, Neurochirurgie (2020), doi: https://doi.org/10.1016/j.neuchi.2019.10.011
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POSITIONAL CRANIAL DEFORMATION IN CHILDREN: A PLEA FOR THE EFFICACY OF THE CRANIAL HELMET IN CHILDREN THIEBAUD
PICART,
MD;
PIERRE-AURELIEN
BEURIAT,
MD;
ALEXANDRU
SZATHMARI, MD, PhD; FEDERICO Di ROCCO, MD, PhD; CARMINE MOTTOLESE, MD, PhD Department of Pediatric Neurosurgery, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Claude Bernard University Lyon 1, 8 Avenue Rockefeller, 69003 Lyon
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Reference center for Craniosynostosis, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Corresponding author: Carmine MOTTOLESE
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Department of Pediatric Neurosurgery
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Hôpital Femme Mère Enfant
32 Avenue du Doyen Jean Lépine, 69677 Bron cedex, France
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Tel: +33 4 72 35 75 72 Fax: + 33 4 72 11 93 28
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E-mail: [email protected]
POSITIONAL CRANIAL DEFORMATION IN CHILDREN: A PLEA FOR THE EFFICACY OF THE CRANIAL HELMET IN CHILDREN
THIEBAUD
PICART,
MD;
PIERRE-AURELIEN
BEURIAT,
MD;
ALEXANDRU
SZATHMARI, MD, PhD; FEDERICO Di ROCCO, M.D, PhD; CARMINE MOTTOLESE, MD, PhD
Department of Pediatric Neurosurgery, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Claude Bernard University Lyon 1, 8 Avenue Rockefeller, 69003 Lyon Reference center for Craniosynostosis, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Corresponding author: Carmine MOTTOLESE
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Department of Pediatric Neurosurgery Hôpital Femme Mère Enfant 32 Avenue du Doyen Jean Lépine, 69677 Bron cedex, France Tel: +33 4 72 35 75 72 Fax: + 33 4 72 11 93 28 E-mail: [email protected]
INTRODUCTION Cranial deformations have historically aroused interest worldwide. Nowadays, positional
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plagiocephaly is a real problem, for parents but also for pediatricians and pediatric neurosurgeons, as incidence is increasing.
The term “plagiocephaly” is derived from the Greek “plagios” (oblique) and “cephalos” (head):
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“oblique head”. The cranial deformity can be anterior or posterior. Generally, plagiocephaly is defined as parallel, because the tangents through the cranial asymmetries in the anterior and in the
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posterior contralateral deformed regions do not intersect.
The term brachycephaly designates a postural posterior cranial deformation that is characterized by
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short anterior–posterior cranial diameter with decreased length, increased transversal diameter and bilateral prominent parietal bossing.
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The cranial sutures are open and the deformity is thus distinct from frontal or posterior plagiocephaly related to a premature closure of the coronal or lambdoid sutures, which constitutes true craniosynostosis.
Positional plagiocephaly is not associated with abnormal cerebral and neuropsychological development. [1] [2]
In 1992 the American Association of Pediatrics recommended that the ventral position should be avoided to prevent the Sudden Infant Death Syndrome (SIDS); however, the preferred supine position resulted in an increased incidence of positional cranial deformation. [3]. Certainly, supine sleep has reduced the number of sudden deaths in children by more than 40%, but has increased the rate of cranial deformation [3] and also the importance of the esthetic consequences. As reported by Piatt at al. in 2004, problems concerning the shape of a child’s head are a frequent reason for consulting a pediatric neurosurgeon [4, 5][6, 7].
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The real incidence of positional plagiocephaly is difficult to establish, because the number of pediatric out-patient consultations for this problem has greatly increased. Incidence ranges between 1 in 60 and 1 in 3,500 live births [8–11]. Boere Bonekamp et al. reported incidence of 8.2%, with variations according to age, with a first peak at 16% at 6 weeks, increasing to 19.7% at 4 months and then declining to an incidence of 3% [12]. According to Martinez-Lage et al., incidence ranges between 18% and 20 % in children born in good health and diminishes with age, highlighting the fact that some cases improve spontaneously [13]. Postnatal cranial flattening is found in 56% of multiple births and only 13% of single births [14]. Kane reported overall incidence of 48% in healthy infants younger than 1 year, depending on the
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sensitivity of the diagnostic criteria. [7]. Other investigators reported that the incidence of positional plagiocephaly is almost 50% in infants under 3 months of age [15].
In contrast to the high incidence of deformational plagiocephaly, it is important to remember that
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true lambdoid synostotic plagiocephaly is very rare, with incidence of 3 in 100,000 births (0.003%), according to Rekate et al. [9].
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A debated point about positional plagiocephaly concerns choice of treatment: some authors fear the impact of the cranial deformation on the growing brain, while others reported that cranial and facial
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asymmetries improve over time and that the problem is merely esthetic [7, 16–21]. It is often said that deformational plagiocephaly has no impact on brain development and is only an
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esthetic issue [18, 19]. The aim of treatment is only to improve esthetics and prevent possible ocular, respiratory or swallowing dysfunction related to the facial deformation. Others series report neurological disorders: cognitive or motor retardation, visual field restriction, and psychological stress due to bullying and to muscular problems [5, 6, 18, 22–24]. Infants and pre-school children with deformational plagiocephaly scored lower on developmental tests than children without skull deformity, but not significantly [25, 26]. Treatment by cranial orthosis or helmet is generally adopted in many countries all over the world and is based on the concept of applying forces on the unaffected skull so as to orientate cranial growth through the deformed region and promote correction (Figure 1). We report our experience with cranial orthoses in positional cranial deformation. MATERIAL AND METHODS A retrospective systematic review was conducted of 2,188 patients (75% male, 25% female) with positional head deformity treated by cranial helmet between 1991 and 2013. Data comprised 3
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gender, date of birth, type of plagiocephaly (bilateral or asymmetric, and laterality) date of beginning and end of treatment, and morphologic parameters at baseline and after treatment: head circumference, right and left tragus to lateral canthus distance, right and left tragus to corner of the mouth distance, anteroposterior diameter, lateral diameter, cranial index and cranial diagonals difference (CDD). Children presenting unilateral deformation plagiocephaly (UDP) were classified into 3 groups according to severity: -
Mild: CDD < 5 mm
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Moderate: 5 mm < CDD < 10 mm
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Severe: CDD > 10 mm
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Then, measures were analyzed to assess the effectiveness and tolerance of the cranial helmet: Restoration of facial symmetry, indicating that treatment was successful, independently of type of plagiocephaly; -
Requirement for posterior cranial remodeling, indicating that treatment failed,
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independently of type of plagiocephaly;
Significant decrease in cranial index, indicating that treatment was successful in the
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brachycephaly subgroup;
Significant decrease in CDD, indicating that treatment was successful in the UDP subgroup.
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Age outset of treatment was available for 2,034 children, and ranged from 35 days to 26.5 months, for a mean 8 months 13 days and a median 8 months 4 days. Age at end of treatment was available for 1,961 children, and ranged from 4 months 22 days to 41 months, for a mean 14 months.
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539 children were treated for brachycephaly: 152 girls (28.2%) and 387 boys (71.8%). Age
outset of treatment was 8 months 17 days. Of the 1,520 children with UDP, 374 were girls (24.6%), 1,146 boys (75.4%). Parietal
flattening was lateralized on the right in 817 cases (53.8%) and on the left in 703 (46.2%). Age outset of treatment was available for 1,514 children, with an average of 8 months 11 days. 52 couples of twin brothers or sisters comprised 4.75% of the population: 27 twins with
brachycephaly and 77 with UDP.
Mean treatment time was 199 days, for a median 186 days (range, 50-667 days). RESULTS Details of the cranial perimeter during treatment are reported in Table 1. At end of treatment, average head circumferences were very similar between groups: within 1 SD according to the WHO data.
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Facial symmetry was considered restored when the right and left distances from the tragus to the lateral canthus and to the corner of the mouth were equal. Facial symmetry pre-existed treatment in 13.7% of children. The rate was significantly higher at end of treatment, at 66.7% (p<0.01), thanks to the effectiveness of the helmet. In the brachycephaly subgroup, the rate of pretreatment facial symmetry was higher, at 40.1%., and significantly increased at end of treatment, at 78.5% (p<0.01), compared to only 4.2% and 62.4% respectively (p<0.01) in the UDP subgroup (Table 2). To assess the effectiveness of the helmet in bilateral plagiocephaly, we compared cranial index at beginning and end of treatment. Brachycephaly is diagnosed for indices >80%. Cranial
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index ranged between 94.4% and 124.2% before treatment and between 86.8% and 121.4% after, for a mean 103.5 ± 6% and 96.7 ± 7.2% respectively (p<0.01): i.e., a considerable improvement (Table 3).
To assess the effectiveness of the helmet in the UDP subgroup, CDD was the most relevant
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parameter. Before treatment, CDD ranged between 0.3 cm and 4.5 cm, for a mean 1.50 ± 0.54 cm. At end of treatment, CDD ranged between 0.1 cm and 2.5 cm, with a significantly lower mean of
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0.72 ± 0.37 cm (p<0.01) (Table 4a).
At beginning of treatment, 2.5% of children presented mild plagiocephaly, 19.6% moderate
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plagiocephaly and 77.9% severe plagiocephaly. At end of treatment, UDP was mild in 40.2% of children, moderate in 44.3% and severe in 15.5%. There were significantly fewer children in the severe subgroups (p<0.01) (Table 4b)
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Surgical management can be proposed, but is rare after orthotic treatment. In only 8 cases
(0.2%), helmet therapy did not achieve good clinical results and surgical posterior cranial remodeling was performed: 3 cases of brachycephaly and 5 of UDP, all for esthetic impact considered too severe by the parents.
Helmet tolerance was very good. Side-effects are reported in Table 5, and were particularly
low in this series. There were skin lesions such as erythema (1.6%) or dermabrasion (0.6%). More severe complications such as osteitis were very rare (0.1%). Nevertheless, these results require very careful oversight by both parents and care-providers to ensure this low rate of skin complications. DISCUSSION The helmet technique was first used in the USA by Clarren et al. in 1979 [27] and we adopted it in 1989. It is generally agreed that positional plagiocephaly can result from external pressure on a rapidly developing skull [9]. The particular malleability of the cranial bones in the fetal period and first
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months of life predisposes to cranial deformation. As reported by Miller and Clarren, associated neck muscle system anomalies prevent symmetrical head rotation, increasing pressure on one side of the skull, counterbalancing the expansion force of the brain on this side and resulting in cranial asymmetry [6]. The intensity and duration of external forces along with rapid cranial growth in the first months of life can explain the different degrees of deformation severity and the peak incidence reported in the first 4 or 6 months of life. This period corresponds to the acquisition of head maintenance and of the sitting position, which can help spontaneous correction of the cranial deformation by avoiding direct application of forces on the skull.
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Risk factors for positional plagiocephaly have been identified [28]: premature birth
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hypotonic condition related to muscle disorder
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congenital torticollis
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intrauterine factors such as multiple pregnancy or oligohydramnios
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bifid uterus
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macrocephaly.
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Postnatal risk factors comprise:
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one side preferred by the newborn for sleep, without particular reason
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contraction of the sternocleidomastoid or trapezius muscle
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4th-nerve palsy inducing head malpositioning to avoid diplopia neurological condition with hypotonic attitude and impaired mobility.
Neuropsychological impairment is not usually associated. Esthetic impairment of facial symmetry can be observed in mild forms while more severe problems can arise in more severe forms: position of orbits, maxilla, nose and jaws, and impact on visual, breathing and swallowing functions.
Treatment duration varies according to patient age, severity of asymmetry, the correction induced, and parental adherence. One of the important factors in helmet effectiveness is age at treatment initiation. Helmet therapy is theoretically indicated in children older than 6 months, suffering from severe plagiocephaly (CDD > 1 cm) [10, 22, 29–32], possibly after failure of other methods [16, 32, 33],
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but, as mentioned above, in severe forms treatment can be early, at 4 months, often in our experience with shorter duration. Because of growth dynamics, the outcome of helmet therapy depends on the timing of treatment initiation, and results may be significantly poorer if treatment starts too late. [10, 22, 34–36] As expected decrease in CDD is 65% before but 51% after 8 months of age [10], earlier initiation is associated with greater efficacy. Advanced age is a risk factor for helmet therapy failure (RR = 1.13) [37]. Efficacy was zero when treatment began after 12 months of age [10, 17]; at this age, the skull is harder and less accessible to the remodeling forces of the helmet, and consequently we do not then propose helmet therapy. The old dogma, well known in pediatrics, held that, between the age of 4 months and 6 months, when children are able to maintain their head
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and acquire the sitting position, spontaneous remodeling restores acceptable cranial shape. Our own experience showed that, when a child presents moderate or severe plagiocephaly at the age of 5 or 6 months, the dysmorphia does not spontaneously recover and treatment by cranial orthosis may be advocated [38].
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Mean time from diagnosis to consultation with a craniofacial specialist is 3.3 months; a longer interval may impair the outcome of helmet therapy. [22] Moreover, more than half the
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children receive additional treatment, delaying the beginning of helmet therapy, which can waste precious time and result in poorer outcome. [22] The decision to start helmet therapy is made by
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parents. This choice is related to their dissatisfaction with their child’s head shape and to the effect they expect from helmet therapy [5]. Treatment durations in the literature are relatively short, between 13 weeks and 5.7 months [10, 16, 19, 29], but were longer in the present series to make
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best use of brain and skull growth, which may explain the high rate of good results in our experience. Efficacy correlates with length of treatment, but the earlier treatment starts, the better the results, because the bone is less rigid and consequently more sensitive to the action of the helmet. In our experience, results are better when treatment is early, before the age of 11-12 months; in clear severe deformation, we recommend starting treatment at 4 months. There are different types of cranial remodeling orthosis, but in France “Starband” is the most commonly used (Figure 2). It can be worn by children from 3 months to 18 months of age. The shape of the helmet is tailored to the correction objectives. Cranial remodeling orthoses are custommade (Figure 3) and require precise molding of the child’s cranium followed by vacuum thermoforming. Experience shows that the helmet does not impede cranial growth, and can be used in any child, including hydrocephalic patients treated with a ventriculo-peritoneal shunt. The inside of the device is coated with foam so as to be as comfortable as possible. The pressure points where forces are
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delivered are selected by the neurosurgeon. Good team-work between neurosurgeon, physiotherapist and helmet manufacturer is essential, to obtain perfect contact between the helmet and the points on the skull where the forces are be exerted. Treatment is always supervised by a neurosurgeon, in liaison with the technician in charge of adjustment. Setting up treatment is timeconsuming. Our follow-up protocol (Table 6) involves patient consultations, with the neurosurgeons working with physiotherapists and helmet manufacturers to program changes in pressure points to adapt the orthosis to morphological changes and growth: this is the principle of dynamic orthotics. The involvement of pediatric neurosurgeons is important to avoid misuse of the orthotic treatment,
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as can often happen without clinical follow-up to reduce the risk of overlooking true craniostenosis, incurring functional risk.
Side-effects, and particularly skin erythema or wounds, should be carefully monitored, first by the
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parents and also by the physiotherapist and neurosurgeons in consultation. To prevent cutaneous lesions, skin massages with ointment are recommended 3 times a day.
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Assessing the severity of cranial asymmetry is important for therapeutic decision-making. In 2004, Argenta et al. classified morphological asymmetry in 5 stages according the degree of cranial
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modifications in positional posterior parallel plagiocephaly or in positional brachycephaly [39]. Measurements are made at the glabella and external occipital protuberance (opistocranion). These
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two points can also indicate the length of the cranium and the distance between the two external acoustic canals.
The brachycephaly is classified in 3 types. Argenta’s classification is useful for deformity severity, but only parental acceptance of their child’s asymmetry can determine treatment. Nevertheless, none of the cranial measurements used estimate esthetic severity, which is obviously subjective.
Radiological assessment of positional plagiocephaly is of little interest because simple cranial Xrays do not show the asymmetry. CT can be more useful to study cranial and basal asymmetry and also the asymmetry of the face, but the problem of irradiation for a growing patient limit its use to when true craniosynostosis is suspected [40, 41]. The need for anesthesia in patients older than 3 months is also a strong argument to forego radiological examination. Ultrasound has been used by some investigators, mainly to monitor sutures progression [42].
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In the last 10 years, infrared laser-assisted scansion of the skull in children has enabled monitoring of cranial modifications, providing an imprint of the skull at the beginning and of cranial development with or without treatment. Our experience confirmed the classical male predominance [16] of plagiocephaly, with 75% boys in our population. This could be explained by hormonal status in fetuses. Testosterone may accentuate muscular action in male fetuses, leading to torticollis, whereas relaxing hormones may loosen female connective tissues. [10, 22, 43] As already reported, multiple pregnancy favors plagiocephaly [10]. The large number of twins in our population (4.75%, versus 1% in the general population) supports this hypothesis (Figure 4).
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Antenatal ultrasound studies in a small cohort of pregnant women showed prenatal existence of cranial deformity in case of deformational plagiocephaly, suggesting that positional cranial deformation can already exist before birth.
At birth, the small size of the skull and the particular emotional atmosphere related to such
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an important event can explain the low rate of detection of cranial deformation. With growth, the larger skull volume reveals the deformation and eases diagnosis.
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In our experience, helmet therapy is an effective and well-tolerated treatment for plagiocephaly. Contrary to the opinion of some authors [32, 44], it seems particularly appropriate
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for correction of brachycephaly, reducing cranial index by 6.8% on average. In children with UDP, CDD was reduced by 0.78 cm (52%) on average. At end of treatment, in UDP, only 15.5% of
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children still showed severe plagiocephaly, requiring surgical treatment in a very few cases (0.2%). The helmet was also effective for facial asymmetry. The rate of facial symmetry increased from 4.2% at beginning of treatment to 62.4% at the end in brachycephaly, and from 2.5% to 40.2%, from 19.5% to 44.2% and from 17% to 77% in mild, moderate and severe forms of UDP, respectively. We were not able to constitute a control group, as our center adopted helmet treatment and developed the technique a long time ago, rapidly becoming a reference center for parents, who come specifically for that.
In a study comparing helmet therapy versus repositioning therapy, Graham et al. found that
helmet therapy led to a final CDD of 0.71 cm (61%), and was more effective than repositioning therapy. [10] In another non-randomized study, including 128 children with UDP, Kluba et al. also reported that cranial vault asymmetry index was reduced by 68% in children treated with a cranial helmet, versus only 31% in children without treatment. [17] Moreover, helmet therapy favored acquisition of facial symmetry in the present series, as also previously reported [16]. Like other authors [16], we think that helmet therapy does not impair global cranial growth, as the average
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cranial perimeter in our series was within 1 SD according to the WHO data. Consequently, our series confirms that helmet therapy optimizes and orients the orthopedic properties of the brain that are at the origin of skull growth. Efficacy depends on changing the pressure points in line with cranial modifications with skull growth and the ongoing correction obtained. Many other authors also consider that helmet therapy is effective [16–19, 22, 23, 27, 32, 35, 44–61]; even so, a few are skeptical [5, 26]. What is clear from our experience is that the helmet does not alter or impede skull growth. To demonstrate efficacy, serial CT-scans would be needed, to quantify skull progression and the changes in basal angles and cranial index; but this is unacceptable due to the high risk of cranial irradiation in young children.
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Changes in cerebrospinal fluid (CSF) dynamics are important in the development of positional plagiocephaly; according to some authors, increased amounts of CSF in subarachnoid spaces could trigger the skull deformation: i.e., external hydrocephaly could induce distortion of the skull. In true craniosynostosis, the involvement of CSF was reported by Carmel in 1981 [62] and, several
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years later, modifications of the subarachnoid spaces were described by Chadduck [63].
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Sawin, as well as other authors, demonstrated that the subarachnoid spaces of patients with occipital deformation were enlarged compared to controls [64–67].
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The subarachnoid space dilatation is similar to what is observed in idiopathic hydrocephalus, which is associated not with brain atrophy but with increased cranial circumference [64, 68].
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These modifications of subarachnoid spaces could be the primary anomaly that precedes skull deformation, or else the consequence of CSF circulation modifications induced by the modified cranial morphology. [64][69]. Dia et al. consequently suggested the simple term “occipital plagiocephaly”, because the two situations express the same problem and because the persistence of the external pressure could lead to true closure of the suture [8, 69]. These hypotheses give possible explanations for the flattening of the cranial deformation,
but neither explains the high incidence of positional plagiocephaly or the very rare incidence of lambdoid craniosynostosis [8].
The present rate of complications was very low. Side-effects were particularly rare (2.5%) and seldom serious. Moreover, in our experience, such minor complications disappear when treatment stops. In the present series, the incidence of such complications did not seem to be influenced by the type of plagiocephaly, although numbers were too small for comparative statistical analysis. Usually, the combination of plagiocephaly and brachycephaly, contrary to severity of deformation, is considered to correlate with a higher risk of complications [70]. 10
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Complication rate correlates with seasonal variation in temperature. During summertime, adherence to treatment needs more attention, with more frequent rest periods and more attentive skin care (drying and cleansing). For Freudlsperge et al., minor complications were far more frequent during helmet therapy (26.3%): pressure sores (13.7%), erythema (2.9%), skin erosion or infection (4.3%) or defective fitting (5.4%). [70] In some series, all parents declared at least one side-effect, with skin irritation frequency up to 96%.[5, 26] In these series, patients wore their helmet 23 hours a day, which is a little bit longer than recommended in our protocol. However, length of treatment has to be adapted to deformation progression, and generally, in our experience, to have a very good result, treatment duration is longer than that reported by some authors in the United States, which was
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generally 3 to 4 months. Finally, it is well recognized that helmet therapy is particularly safe and well-tolerated [18, 27] and does not negatively influence quality of life and of sleep [5]. In our experience, treatment was never interrupted for skin problems and there were no behavioral problems during follow-up until the age of 6 to 8 years.
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Our experience shows that poor parental compliance is a very important risk factor for helmet therapy failure [17] (RR = 2.42 [37]) and complications are almost always related to parental
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negligence. We are aware that simple and clear explanation of the goal and of the treatment protocol is essential to obtain good clinical results. Close follow-up at treatment outset is also mandatory to
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ensure a low rate of complications. [70] In our center, we are available to parents for any problems, as it is very important to provide an early answer and to react rapidly in case of complications or doubts. Simple explanations to parents who have to withdraw the device due to skin irritation avoid
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rapid worsening of the situation. The best attitude is to avoid the problem in the first place: prevention is essential.
Plagiocephaly prevention is an emerging concept. [5] Some authors suggest using
positioning aids from birth onward [22] and providing perinatal physiotherapy advice to parents to reduce cervical muscular contracture [16, 32, 50]. Measurement of cranial diameter should become an obligatory part of pediatric well-child consultations in the 3rd and 4th months of life. [22] Nutritional advice can be provided concerning the importance of adequate vitamin D intake during pregnancy and infancy. Vitamin D insufficiency during pregnancy is associated with skeletal homeostasis disorder in general and is independently associated with increased risk of skull deformation during infancy. Use of formula milk after birth is also associated with skull deformation [43]. Prevention of positional head deformities can involve physiotherapy [32, 55, 71, 72], osteopathy [50], counter-positioning [73], bedding devices [47] and cervical stretching [74]. Wilbrand et al. reported improvements in cranial shape in 50 children using non-orthotic methods 11
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(stretching exercises and adapted pillows). [32] However, pillows can cause side-effects (bilateral apostasis of the external ears). [32] A recent Australian study showed that, in a group of patients treated with helmet, correction of cranial parameters was better than in the group without helmet treatment. [75] Also, while there is no level 1 evidence of efficacy of helmet treatment, all the studies of anatomomorphological indices confirmed that helmets showed real morphological efficacy [23, 46, 48, 51, 54, 56, 60, 61]. While spontaneous reduction of UDP is reported in 70% cases [16, 18, 25, 48, 76, 77], in our experience severe cases never improved, in complete contradiction with the principle that, with the time and neuropsychological development, spontaneous recovery is always possible.
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After 30 years’ experience, we have learned that helmets can be effective up to the age of 19 months; it is very rare to observe significant improvement in skull shape after this age, and we stop treatment at that point. CONCLUSIONS
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Positional plagiocephaly and brachycephaly are a real problem in our society. The need for good quality of life and to avoid personal psychological problems has incited families and doctors
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to address this problem.
Our experience confirms that the cranial helmet is a simple and well-tolerated treatment
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which provides satisfactory clinical results in positional plagiocephaly. Success requires good teamwork with parents and careful management by orthotist, physiotherapist and physician to avoid
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minor side-effects. Helmet therapy must be applied as early as possible, to take full advantage of cranial bone plasticity. Unfortunately, this alternative treatment remains relatively under-used in France, undoubtedly because few pediatric neurosurgical departments propose it. Nevertheless, there remain many controversies, in particular concerning long-term esthetic and functional outcome. A long-term multicenter prospective study is needed to remove doubts. Finally, “acceptable” head shape in infants and at an older age is subjective. What is certain
is that cranial asymmetry persists into adolescence and adulthood. The psychological consequences and acceptance by parents and child are less certain. As Montaigne said, "It is better to have a well-structured head than a full head" - a wellstructured head with a nice craniofacial appearance! [78].
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FIGURE LEGENDS Figure 1: Diagram of the force applied by the helmet to remodel the head shape. Figure 2 A and B: Pictures of children wearing a helmet Figure 3: 3D measurement for tailored helmet.
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Figure 4: Picture of twins wearing a helmet.
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5. van Wijk RM, van Til JA, Groothuis-Oudshoorn CGM, et al. (2014) Parents’ decision for helmet therapy in infants with skull deformation. Childs Nerv Syst 30:1225–1232. https://doi.org/10.1007/s00381-014-2399-2 6. Miller RI, Clarren SK (2000) Long-term developmental outcomes in patients with deformational plagiocephaly. Pediatrics 105:E26
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7. Kane AA, Mitchell LE, Craven KP, Marsh JL (1996) Observations on a recent increase in plagiocephaly without synostosis. Pediatrics 97:877–885
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8. Dias MS, Klein DM (1996) Occipital plagiocephaly: deformation or lambdoid synostosis? II. A unifying theory regarding pathogenesis. Pediatr Neurosurg 24:69–73. https://doi.org/10.1159/000121019
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9. Rekate HL (1998) Occipital plagiocephaly: a critical review of the literature. J Neurosurg 89:24–30. https://doi.org/10.3171/jns.1998.89.1.0024
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10. Graham JM, Gomez M, Halberg A, et al. (2005) Management of deformational plagiocephaly: repositioning versus orthotic therapy. J Pediatr 146:258–262. https://doi.org/10.1016/j.jpeds.2004.10.016 11. Argenta LC, David LR, Wilson JA, Bell WO (1996) An increase in infant cranial deformity with supine sleeping position. J Craniofac Surg 7:5–11 12. van Wijk RM, van Vlimmeren LA, Groothuis-Oudshoorn CGM, et al. (2014) Helmet therapy in infants with positional skull deformation: randomised controlled trial. BMJ 348:g2741 13. Martínez-Lage JF, Arráez Manrique C, Ruiz-Espejo AM, et al. (2012) Positional cranial deformations: a clinical-epidemiological study. An Pediatr Barc Spain 2003 77:176–183. https://doi.org/10.1016/j.anpedi.2012.02.013 14. Peitsch WK, Keefer CH, LaBrie RA, Mulliken JB (2002) Incidence of cranial asymmetry in healthy newborns. Pediatrics 110:e72 15. Mawji A, Vollman AR, Hatfield J, et al. (2013) The incidence of positional plagiocephaly: a cohort study. Pediatrics 132:298–304. https://doi.org/10.1542/peds.2012-3438 16. Vernet O, de Ribaupierre S, Cavin B, Rilliet B (2008) Treatment of posterior positional plagiocephaly. Arch Pediatr 15:1829–1833. https://doi.org/10.1016/j.arcped.2008.09.007 17. Kluba S, Kraut W, Calgeer B, et al. (2014) Treatment of positional plagiocephaly--helmet or no helmet? J Cranio-Maxillo-Fac Surg 42:683–688. https://doi.org/10.1016/j.jcms.2013.09.015 18.
Flannery ABK, Looman WS, Kemper K (2012) Evidence-based care of the child with
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deformational plagiocephaly, part II: management. J Pediatr Health Care Off 26:320–331. https://doi.org/10.1016/j.pedhc.2011.10.002 19. Gautschi OP, Rilliet B, Schaller K, Jenny B (2013) Positional plagiocephaly in infancy: diagnosis and management. Praxis 102:1537–1542. https://doi.org/10.1024/1661-8157/a001506 20. Speltz ML, Collett BR, Stott-Miller M, et al. (2010) Case-control study of neurodevelopment in deformational plagiocephaly. Pediatrics 125:e537-542. https://doi.org/10.1542/peds.2009-0052 21. Collett B, Breiger D, King D, et al. (2005) Neurodevelopmental implications of “deformational” plagiocephaly. J Dev Behav Pediatr JDBP 26:379–389 22. Kluba S, Lypke J, Kraut W, et al. (2014) Preclinical pathways to treatment in infants with positional cranial deformity. Int J Oral Maxillofac Surg 43:1171–1175. https://doi.org/10.1016/j.ijom.2014.05.011
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23. Lee RP, Teichgraeber JF, Baumgartner JE, et al. (2008) Long-term treatment effectiveness of molding helmet therapy in the correction of posterior deformational plagiocephaly: a five-year followup. Cleft Palate-Craniofacial J 45:240–245. https://doi.org/10.1597/06-210.1 24. Steinbok P, Lam D, Singh S, et al. (2007) Long-term outcome of infants with positional occipital plagiocephaly. Childs Nerv Syst 23:1275–1283. https://doi.org/10.1007/s00381-007-0373-y
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25. Collett BR, Gray KE, Starr JR, et al. (2013) Development at age 36 months in children with deformational plagiocephaly. Pediatrics 131:e109-115. https://doi.org/10.1542/peds.2012-1779
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26. Rowland K, Das N (2015) PURLs: helmets for positional skull deformities: a good idea, or not? J Fam Pract 64:44–46
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27. Clarren SK, Smith DW, Hanson JW (1979) Helmet treatment for plagiocephaly and congenital muscular torticollis. J Pediatr 94:43–46
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28. De Bock F, Braun V, Renz-Polster H (2017) Deformational plagiocephaly in normal infants: a systematic review of causes and hypotheses. Arch Dis Child 102:535–542. https://doi.org/10.1136/archdischild-2016-312018 29. Branch LG, Kesty K, Krebs E, et al. (2015) Argenta clinical classification of deformational plagiocephaly. J Craniofac Surg 26:606–610. https://doi.org/10.1097/SCS.0000000000001511 30. Clarren SK (1981) Plagiocephaly and torticollis: etiology, natural history, and helmet treatment. J Pediatr 98:92–95 31. Laughlin J, Luerssen TG, Dias MS, Committee on Practice and Ambulatory Medicine, Section on Neurological Surgery (2011) Prevention and management of positional skull deformities in infants. Pediatrics 128:1236–1241. https://doi.org/10.1542/peds.2011-2220 32. Wilbrand J-F, Seidl M, Wilbrand M, et al. (2013) A prospective randomized trial on preventative methods for positional head deformity: physiotherapy versus a positioning pillow. J Pediatr 162:1216–1221, 1221.e1. https://doi.org/10.1016/j.jpeds.2012.11.076 33. Biggs WS (2003) Diagnosis and management of positional head deformity. Am Fam Physician 67:1953–1956 34. Kelly KM, Littlefield TR, Pomatto JK, et al. (1999) Importance of early recognition and treatment of deformational plagiocephaly with orthotic cranioplasty. Cleft Palate-Craniofacial J 36:127–130. https://doi.org/10.1597/1545-1569(1999)036<0127:IOERAT>2.3.CO;2 35.
Kluba S, Kraut W, Reinert S, Krimmel M (2011) What is the optimal time to start helmet
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therapy in positional plagiocephaly? Plast Reconstr Surg 128:492–498. https://doi.org/10.1097/PRS.0b013e31821b62d6 36. Thompson JT, David LR, Wood B, et al. (2009) Outcome analysis of helmet therapy for positional plagiocephaly using a three-dimensional surface scanning laser. J Craniofac Surg 20:362– 365. https://doi.org/10.1097/SCS.0b013e3181992382 37. Steinberg JP, Rawlani R, Humphries LS, et al. (2015) Effectiveness of conservative therapy and helmet therapy for positional cranial deformation. Plast Reconstr Surg 135:833–842. https://doi.org/10.1097/PRS.0000000000000955 38. Di Rocco F, Ble V, Beuriat P-A, et al. (2019) Prevalence and severity of positional plagiocephaly in children and adolescents. Acta Neurochir (Wien) 161:1095–1098. https://doi.org/10.1007/s00701-019-03924-2
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39. Argenta L, David L, Thompson J (2004) Clinical classification of positional plagiocephaly. J Craniofac Surg 15:368–372 40. Brenner D, Elliston C, Hall E, Berdon W (2001) Estimated risks of radiation-induced fatal cancer from pediatric CT. AJR Am J Roentgenol 176:289–296. https://doi.org/10.2214/ajr.176.2.1760289
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41. Pearce MS, Salotti JA, Little MP, et al. (2012) Radiation exposure from CT scans in childhood and subsequent risk of leukaemia and brain tumours: a retrospective cohort study. Lancet Lond Engl 380:499–505. https://doi.org/10.1016/S0140-6736(12)60815-0
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42. Sze RW, Parisi MT, Sidhu M, et al. (2003) Ultrasound screening of the lambdoid suture in the child with posterior plagiocephaly. Pediatr Radiol 33:630–636. https://doi.org/10.1007/s00247-0031009-3 43. Weernink MGM, van Wijk RM, Groothuis-Oudshoorn CGM, et al. (2016) Insufficient vitamin D supplement use during pregnancy and early childhood: a risk factor for positional skull deformation. Matern Child Nutr 12:177–188. https://doi.org/10.1111/mcn.12153
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44. Teichgraeber JF, Seymour-Dempsey K, Baumgartner JE, et al. (2004) Molding helmet therapy in the treatment of brachycephaly and plagiocephaly. J Craniofac Surg 15:118–123 45. Bialocerkowski AE, Vladusic SL, Howell SM (2005) Conservative interventions for positional plagiocephaly: a systematic review. Dev Med Child Neurol 47:563–570 46. Couture DE, Crantford JC, Somasundaram A, et al. (2013) Efficacy of passive helmet therapy for deformational plagiocephaly: report of 1050 cases. Neurosurg Focus 35:E4. https://doi.org/10.3171/2013.8.FOCUS13258 47. de Chalain T (2003) The Safe-T-Sleep device: safety and efficacy in maintaining infant sleeping position. N Z Med J 116:U581 48. Goh JL, Bauer DF, Durham SR, Stotland MA (2013) Orthotic (helmet) therapy in the treatment of plagiocephaly. Neurosurg Focus 35:E2. https://doi.org/10.3171/2013.7.FOCUS13260 49. Govaert B, Michels A, Colla C, van der Hulst R (2008) Molding therapy of positional plagiocephaly: subjective outcome and quality of life. J Craniofac Surg 19:56–58. https://doi.org/10.1097/SCS.0b013e31815c8a27 50. Lessard S, Gagnon I, Trottier N (2011) Exploring the impact of osteopathic treatment on cranial asymmetries associated with nonsynostotic plagiocephaly in infants. Complement Ther Clin Pract 17:193–198. https://doi.org/10.1016/j.ctcp.2011.02.001
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51. Lipira AB, Gordon S, Darvann TA, et al. (2010) Helmet versus active repositioning for plagiocephaly: a three-dimensional analysis. Pediatrics 126:e936-945. https://doi.org/10.1542/peds.2009-1249 52. Mortenson P, Steinbok P, Smith D (2012) Deformational plagiocephaly and orthotic treatment: indications and limitations. Childs Nerv Syst 28:1407–1412. https://doi.org/10.1007/s00381-012-1755-3 53. Mulliken JB, Vander Woude DL, Hansen M, et al. (1999) Analysis of posterior plagiocephaly: deformational versus synostotic. Plast Reconstr Surg 103:371–380 54. Naidoo SD, Skolnick GB, Patel KB, et al. (2015) Long-term outcomes in treatment of deformational plagiocephaly and brachycephaly using helmet therapy and repositioning: a longitudinal cohort study. Childs Nerv Syst 31:1547–1552. https://doi.org/10.1007/s00381-015-2769-4
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55. Neufeld S, Birkett S (2000) What to do about flat heads: preventing and treating positional occipital flattening. Axone Dartm NS 22:29–31 56. Plank LH, Giavedoni B, Lombardo JR, et al. (2006) Comparison of infant head shape changes in deformational plagiocephaly following treatment with a cranial remolding orthosis using a noninvasive laser shape digitizer. J Craniofac Surg 17:1084–1091. https://doi.org/10.1097/01.scs.0000244920.07383.85
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57. Robinson S, Proctor M (2009) Diagnosis and management of deformational plagiocephaly. J Neurosurg Pediatr 3:284–295. https://doi.org/10.3171/2009.1.PEDS08330
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58. Rogers GF, Miller J, Mulliken JB (2008) Comparison of a modifiable cranial cup versus repositioning and cervical stretching for the early correction of deformational posterior plagiocephaly. Plast Reconstr Surg 121:941–947. https://doi.org/10.1097/01.prs.0000299938.00229.3e 59. Vles JS, Colla C, Weber JW, et al. (2000) Helmet versus nonhelmet treatment in nonsynostotic positional posterior plagiocephaly. J Craniofac Surg 11:572–574
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60. Xia JJ, Kennedy KA, Teichgraeber JF, et al. (2008) Nonsurgical treatment of deformational plagiocephaly: a systematic review. Arch Pediatr Adolesc Med 162:719–727. https://doi.org/10.1001/archpedi.162.8.719 61. Yoo H-S, Rah DK, Kim YO (2012) Outcome analysis of cranial molding therapy in nonsynostotic plagiocephaly. Arch Plast Surg 39:338–344. https://doi.org/10.5999/aps.2012.39.4.338 62. Carmel PW, Luken MG, Ascherl GF (1981) Craniosynostosis: computed tomographic evaluation of skull base and calvarial deformities and associated intracranial changes. Neurosurgery 9:366–372 63. Chadduck WM, Chadduck JB, Boop FA (1992) The subarachnoid spaces in craniosynostosis. Neurosurgery 30:867–871 64. Sawin PD, Muhonen MG, Menezes AH (1996) Quantitative analysis of cerebrospinal fluid spaces in children with occipital plagiocephaly. J Neurosurg 85:428–434. https://doi.org/10.3171/jns.1996.85.3.0428 65. Thaller SR, Hoyt J, Boggan J (1992) Surgical correction of unilateral lambdoid synostosis: occipital rotation flap. J Craniofac Surg 3:12–17; discussion 18-19 66. Muakkassa KF, Hoffman HJ, Hinton DR, et al. (1984) Lambdoid synostosis. Part 2: Review of cases managed at The Hospital for Sick Children, 1972-1982. J Neurosurg 61:340–347. https://doi.org/10.3171/jns.1984.61.2.0340
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67. Fernbach SK, Feinstein KA (1991) Radiologic evaluation of the child with craniosynostosis. Neurosurg Clin N Am 2:569–585 68. Hamza M, Bodensteiner JB, Noorani PA, Barnes PD (1987) Benign extracerebral fluid collections: a cause of macrocrania in infancy. Pediatr Neurol 3:218–221 69. Hinton DR, Becker LE, Muakkassa KF, Hoffman HJ (1984) Lambdoid synostosis. Part 1. The lambdoid suture: normal development and pathology of “synostosis.” J Neurosurg 61:333–339. https://doi.org/10.3171/jns.1984.61.2.0333 70. Freudlsperger C, Bodem JP, Kargus S, et al. (2015) The Incidence of Complications Associated With Molding Helmet Therapy: An Avoidable Risk in the Treatment of Positional Head Deformities? J Craniofac Surg 26:e299-302. https://doi.org/10.1097/SCS.0000000000001649
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71. van Vlimmeren LA, van der Graaf Y, Boere-Boonekamp MM, et al. (2008) Effect of pediatric physical therapy on deformational plagiocephaly in children with positional preference: a randomized controlled trial. Arch Pediatr Adolesc Med 162:712–718. https://doi.org/10.1001/archpedi.162.8.712 72. Vargish L, Mendoza MD, Ewigman B (2009) Use physical therapy to head off this deformity in infants. Consider early PT to prevent severe deformational plagiocephaly. J Fam Pract 58:E1-3
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74. Morrison CS, Chariker M (2006) Positional plagiocephaly: pathogenesis, diagnosis, and management. J Ky Med Assoc 104:136–140 75. Ho JPK, Mallitt K-A, Jacobson E, Reddy R (2016) Use of external orthotic helmet therapy in positional plagiocephaly. J Clin Neurosci 29:46–51. https://doi.org/10.1016/j.jocn.2015.12.023
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76. Hutchison BL, Hutchison LAD, Thompson JMD, Mitchell EA (2004) Plagiocephaly and brachycephaly in the first two years of life: a prospective cohort study. Pediatrics 114:970–980. https://doi.org/10.1542/peds.2003-0668-F
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77. Jones BM, Hayward R, Evans R, Britto J (1997) Occipital plagiocephaly: an epidemic of craniosynostosis? BMJ 315:693–694 78. Mottolese C, Szathmari A, Ricci AC, et al. (2006) Positional plagiocephaly: the place of cranial orthotics. Neurochirurgie 52:184–194
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PII:
S0028-3770(20)30001-1
DOI:
https://doi.org/doi:10.1016/j.neuchi.2019.10.011
Reference:
NEUCHI 1050
To appear in:
Neurochirurgie
Please cite this article as: Picart T, Beuriat P-Aurelien, Szathmari A, Rocco FD, Mottolese C, Positional Cranial Deformation In Children: A Plea For The Efficacy Of The Cranial Helmet In Children, Neurochirurgie (2020), doi: https://doi.org/10.1016/j.neuchi.2019.10.011
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POSITIONAL CRANIAL DEFORMATION IN CHILDREN: A PLEA FOR THE EFFICACY OF THE CRANIAL HELMET IN CHILDREN THIEBAUD
PICART,
MD;
PIERRE-AURELIEN
BEURIAT,
MD;
ALEXANDRU
SZATHMARI, MD, PhD; FEDERICO Di ROCCO, MD, PhD; CARMINE MOTTOLESE, MD, PhD Department of Pediatric Neurosurgery, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Claude Bernard University Lyon 1, 8 Avenue Rockefeller, 69003 Lyon
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Reference center for Craniosynostosis, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Corresponding author: Carmine MOTTOLESE
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Department of Pediatric Neurosurgery
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Hôpital Femme Mère Enfant
32 Avenue du Doyen Jean Lépine, 69677 Bron cedex, France
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Tel: +33 4 72 35 75 72 Fax: + 33 4 72 11 93 28
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E-mail: [email protected]
POSITIONAL CRANIAL DEFORMATION IN CHILDREN: A PLEA FOR THE EFFICACY OF THE CRANIAL HELMET IN CHILDREN
THIEBAUD
PICART,
MD;
PIERRE-AURELIEN
BEURIAT,
MD;
ALEXANDRU
SZATHMARI, MD, PhD; FEDERICO Di ROCCO, M.D, PhD; CARMINE MOTTOLESE, MD, PhD
Department of Pediatric Neurosurgery, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Claude Bernard University Lyon 1, 8 Avenue Rockefeller, 69003 Lyon Reference center for Craniosynostosis, Hôpital Femme Mère Enfant, 32 Avenue du Doyen Jean Lépine, 69677 Bron Cedex, France Corresponding author: Carmine MOTTOLESE
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Department of Pediatric Neurosurgery Hôpital Femme Mère Enfant 32 Avenue du Doyen Jean Lépine, 69677 Bron cedex, France Tel: +33 4 72 35 75 72 Fax: + 33 4 72 11 93 28 E-mail: [email protected]
INTRODUCTION Cranial deformations have historically aroused interest worldwide. Nowadays, positional
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plagiocephaly is a real problem, for parents but also for pediatricians and pediatric neurosurgeons, as incidence is increasing.
The term “plagiocephaly” is derived from the Greek “plagios” (oblique) and “cephalos” (head):
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“oblique head”. The cranial deformity can be anterior or posterior. Generally, plagiocephaly is defined as parallel, because the tangents through the cranial asymmetries in the anterior and in the
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posterior contralateral deformed regions do not intersect.
The term brachycephaly designates a postural posterior cranial deformation that is characterized by
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short anterior–posterior cranial diameter with decreased length, increased transversal diameter and bilateral prominent parietal bossing.
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The cranial sutures are open and the deformity is thus distinct from frontal or posterior plagiocephaly related to a premature closure of the coronal or lambdoid sutures, which constitutes true craniosynostosis.
Positional plagiocephaly is not associated with abnormal cerebral and neuropsychological development. [1] [2]
In 1992 the American Association of Pediatrics recommended that the ventral position should be avoided to prevent the Sudden Infant Death Syndrome (SIDS); however, the preferred supine position resulted in an increased incidence of positional cranial deformation. [3]. Certainly, supine sleep has reduced the number of sudden deaths in children by more than 40%, but has increased the rate of cranial deformation [3] and also the importance of the esthetic consequences. As reported by Piatt at al. in 2004, problems concerning the shape of a child’s head are a frequent reason for consulting a pediatric neurosurgeon [4, 5][6, 7].
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The real incidence of positional plagiocephaly is difficult to establish, because the number of pediatric out-patient consultations for this problem has greatly increased. Incidence ranges between 1 in 60 and 1 in 3,500 live births [8–11]. Boere Bonekamp et al. reported incidence of 8.2%, with variations according to age, with a first peak at 16% at 6 weeks, increasing to 19.7% at 4 months and then declining to an incidence of 3% [12]. According to Martinez-Lage et al., incidence ranges between 18% and 20 % in children born in good health and diminishes with age, highlighting the fact that some cases improve spontaneously [13]. Postnatal cranial flattening is found in 56% of multiple births and only 13% of single births [14]. Kane reported overall incidence of 48% in healthy infants younger than 1 year, depending on the
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sensitivity of the diagnostic criteria. [7]. Other investigators reported that the incidence of positional plagiocephaly is almost 50% in infants under 3 months of age [15].
In contrast to the high incidence of deformational plagiocephaly, it is important to remember that
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true lambdoid synostotic plagiocephaly is very rare, with incidence of 3 in 100,000 births (0.003%), according to Rekate et al. [9].
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A debated point about positional plagiocephaly concerns choice of treatment: some authors fear the impact of the cranial deformation on the growing brain, while others reported that cranial and facial
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asymmetries improve over time and that the problem is merely esthetic [7, 16–21]. It is often said that deformational plagiocephaly has no impact on brain development and is only an
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esthetic issue [18, 19]. The aim of treatment is only to improve esthetics and prevent possible ocular, respiratory or swallowing dysfunction related to the facial deformation. Others series report neurological disorders: cognitive or motor retardation, visual field restriction, and psychological stress due to bullying and to muscular problems [5, 6, 18, 22–24]. Infants and pre-school children with deformational plagiocephaly scored lower on developmental tests than children without skull deformity, but not significantly [25, 26]. Treatment by cranial orthosis or helmet is generally adopted in many countries all over the world and is based on the concept of applying forces on the unaffected skull so as to orientate cranial growth through the deformed region and promote correction (Figure 1). We report our experience with cranial orthoses in positional cranial deformation. MATERIAL AND METHODS A retrospective systematic review was conducted of 2,188 patients (75% male, 25% female) with positional head deformity treated by cranial helmet between 1991 and 2013. Data comprised 3
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gender, date of birth, type of plagiocephaly (bilateral or asymmetric, and laterality) date of beginning and end of treatment, and morphologic parameters at baseline and after treatment: head circumference, right and left tragus to lateral canthus distance, right and left tragus to corner of the mouth distance, anteroposterior diameter, lateral diameter, cranial index and cranial diagonals difference (CDD). Children presenting unilateral deformation plagiocephaly (UDP) were classified into 3 groups according to severity: -
Mild: CDD < 5 mm
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Moderate: 5 mm < CDD < 10 mm
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Severe: CDD > 10 mm
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Then, measures were analyzed to assess the effectiveness and tolerance of the cranial helmet: Restoration of facial symmetry, indicating that treatment was successful, independently of type of plagiocephaly; -
Requirement for posterior cranial remodeling, indicating that treatment failed,
-
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independently of type of plagiocephaly;
Significant decrease in cranial index, indicating that treatment was successful in the
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brachycephaly subgroup;
Significant decrease in CDD, indicating that treatment was successful in the UDP subgroup.
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Age outset of treatment was available for 2,034 children, and ranged from 35 days to 26.5 months, for a mean 8 months 13 days and a median 8 months 4 days. Age at end of treatment was available for 1,961 children, and ranged from 4 months 22 days to 41 months, for a mean 14 months.
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539 children were treated for brachycephaly: 152 girls (28.2%) and 387 boys (71.8%). Age
outset of treatment was 8 months 17 days. Of the 1,520 children with UDP, 374 were girls (24.6%), 1,146 boys (75.4%). Parietal
flattening was lateralized on the right in 817 cases (53.8%) and on the left in 703 (46.2%). Age outset of treatment was available for 1,514 children, with an average of 8 months 11 days. 52 couples of twin brothers or sisters comprised 4.75% of the population: 27 twins with
brachycephaly and 77 with UDP.
Mean treatment time was 199 days, for a median 186 days (range, 50-667 days). RESULTS Details of the cranial perimeter during treatment are reported in Table 1. At end of treatment, average head circumferences were very similar between groups: within 1 SD according to the WHO data.
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Facial symmetry was considered restored when the right and left distances from the tragus to the lateral canthus and to the corner of the mouth were equal. Facial symmetry pre-existed treatment in 13.7% of children. The rate was significantly higher at end of treatment, at 66.7% (p<0.01), thanks to the effectiveness of the helmet. In the brachycephaly subgroup, the rate of pretreatment facial symmetry was higher, at 40.1%., and significantly increased at end of treatment, at 78.5% (p<0.01), compared to only 4.2% and 62.4% respectively (p<0.01) in the UDP subgroup (Table 2). To assess the effectiveness of the helmet in bilateral plagiocephaly, we compared cranial index at beginning and end of treatment. Brachycephaly is diagnosed for indices >80%. Cranial
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index ranged between 94.4% and 124.2% before treatment and between 86.8% and 121.4% after, for a mean 103.5 ± 6% and 96.7 ± 7.2% respectively (p<0.01): i.e., a considerable improvement (Table 3).
To assess the effectiveness of the helmet in the UDP subgroup, CDD was the most relevant
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parameter. Before treatment, CDD ranged between 0.3 cm and 4.5 cm, for a mean 1.50 ± 0.54 cm. At end of treatment, CDD ranged between 0.1 cm and 2.5 cm, with a significantly lower mean of
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0.72 ± 0.37 cm (p<0.01) (Table 4a).
At beginning of treatment, 2.5% of children presented mild plagiocephaly, 19.6% moderate
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plagiocephaly and 77.9% severe plagiocephaly. At end of treatment, UDP was mild in 40.2% of children, moderate in 44.3% and severe in 15.5%. There were significantly fewer children in the severe subgroups (p<0.01) (Table 4b)
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Surgical management can be proposed, but is rare after orthotic treatment. In only 8 cases
(0.2%), helmet therapy did not achieve good clinical results and surgical posterior cranial remodeling was performed: 3 cases of brachycephaly and 5 of UDP, all for esthetic impact considered too severe by the parents.
Helmet tolerance was very good. Side-effects are reported in Table 5, and were particularly
low in this series. There were skin lesions such as erythema (1.6%) or dermabrasion (0.6%). More severe complications such as osteitis were very rare (0.1%). Nevertheless, these results require very careful oversight by both parents and care-providers to ensure this low rate of skin complications. DISCUSSION The helmet technique was first used in the USA by Clarren et al. in 1979 [27] and we adopted it in 1989. It is generally agreed that positional plagiocephaly can result from external pressure on a rapidly developing skull [9]. The particular malleability of the cranial bones in the fetal period and first
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months of life predisposes to cranial deformation. As reported by Miller and Clarren, associated neck muscle system anomalies prevent symmetrical head rotation, increasing pressure on one side of the skull, counterbalancing the expansion force of the brain on this side and resulting in cranial asymmetry [6]. The intensity and duration of external forces along with rapid cranial growth in the first months of life can explain the different degrees of deformation severity and the peak incidence reported in the first 4 or 6 months of life. This period corresponds to the acquisition of head maintenance and of the sitting position, which can help spontaneous correction of the cranial deformation by avoiding direct application of forces on the skull.
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Risk factors for positional plagiocephaly have been identified [28]: premature birth
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hypotonic condition related to muscle disorder
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congenital torticollis
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intrauterine factors such as multiple pregnancy or oligohydramnios
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bifid uterus
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macrocephaly.
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Postnatal risk factors comprise:
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-
one side preferred by the newborn for sleep, without particular reason
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contraction of the sternocleidomastoid or trapezius muscle
-
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-
4th-nerve palsy inducing head malpositioning to avoid diplopia neurological condition with hypotonic attitude and impaired mobility.
Neuropsychological impairment is not usually associated. Esthetic impairment of facial symmetry can be observed in mild forms while more severe problems can arise in more severe forms: position of orbits, maxilla, nose and jaws, and impact on visual, breathing and swallowing functions.
Treatment duration varies according to patient age, severity of asymmetry, the correction induced, and parental adherence. One of the important factors in helmet effectiveness is age at treatment initiation. Helmet therapy is theoretically indicated in children older than 6 months, suffering from severe plagiocephaly (CDD > 1 cm) [10, 22, 29–32], possibly after failure of other methods [16, 32, 33],
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but, as mentioned above, in severe forms treatment can be early, at 4 months, often in our experience with shorter duration. Because of growth dynamics, the outcome of helmet therapy depends on the timing of treatment initiation, and results may be significantly poorer if treatment starts too late. [10, 22, 34–36] As expected decrease in CDD is 65% before but 51% after 8 months of age [10], earlier initiation is associated with greater efficacy. Advanced age is a risk factor for helmet therapy failure (RR = 1.13) [37]. Efficacy was zero when treatment began after 12 months of age [10, 17]; at this age, the skull is harder and less accessible to the remodeling forces of the helmet, and consequently we do not then propose helmet therapy. The old dogma, well known in pediatrics, held that, between the age of 4 months and 6 months, when children are able to maintain their head
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and acquire the sitting position, spontaneous remodeling restores acceptable cranial shape. Our own experience showed that, when a child presents moderate or severe plagiocephaly at the age of 5 or 6 months, the dysmorphia does not spontaneously recover and treatment by cranial orthosis may be advocated [38].
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Mean time from diagnosis to consultation with a craniofacial specialist is 3.3 months; a longer interval may impair the outcome of helmet therapy. [22] Moreover, more than half the
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children receive additional treatment, delaying the beginning of helmet therapy, which can waste precious time and result in poorer outcome. [22] The decision to start helmet therapy is made by
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parents. This choice is related to their dissatisfaction with their child’s head shape and to the effect they expect from helmet therapy [5]. Treatment durations in the literature are relatively short, between 13 weeks and 5.7 months [10, 16, 19, 29], but were longer in the present series to make
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best use of brain and skull growth, which may explain the high rate of good results in our experience. Efficacy correlates with length of treatment, but the earlier treatment starts, the better the results, because the bone is less rigid and consequently more sensitive to the action of the helmet. In our experience, results are better when treatment is early, before the age of 11-12 months; in clear severe deformation, we recommend starting treatment at 4 months. There are different types of cranial remodeling orthosis, but in France “Starband” is the most commonly used (Figure 2). It can be worn by children from 3 months to 18 months of age. The shape of the helmet is tailored to the correction objectives. Cranial remodeling orthoses are custommade (Figure 3) and require precise molding of the child’s cranium followed by vacuum thermoforming. Experience shows that the helmet does not impede cranial growth, and can be used in any child, including hydrocephalic patients treated with a ventriculo-peritoneal shunt. The inside of the device is coated with foam so as to be as comfortable as possible. The pressure points where forces are
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delivered are selected by the neurosurgeon. Good team-work between neurosurgeon, physiotherapist and helmet manufacturer is essential, to obtain perfect contact between the helmet and the points on the skull where the forces are be exerted. Treatment is always supervised by a neurosurgeon, in liaison with the technician in charge of adjustment. Setting up treatment is timeconsuming. Our follow-up protocol (Table 6) involves patient consultations, with the neurosurgeons working with physiotherapists and helmet manufacturers to program changes in pressure points to adapt the orthosis to morphological changes and growth: this is the principle of dynamic orthotics. The involvement of pediatric neurosurgeons is important to avoid misuse of the orthotic treatment,
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as can often happen without clinical follow-up to reduce the risk of overlooking true craniostenosis, incurring functional risk.
Side-effects, and particularly skin erythema or wounds, should be carefully monitored, first by the
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parents and also by the physiotherapist and neurosurgeons in consultation. To prevent cutaneous lesions, skin massages with ointment are recommended 3 times a day.
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Assessing the severity of cranial asymmetry is important for therapeutic decision-making. In 2004, Argenta et al. classified morphological asymmetry in 5 stages according the degree of cranial
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modifications in positional posterior parallel plagiocephaly or in positional brachycephaly [39]. Measurements are made at the glabella and external occipital protuberance (opistocranion). These
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two points can also indicate the length of the cranium and the distance between the two external acoustic canals.
The brachycephaly is classified in 3 types. Argenta’s classification is useful for deformity severity, but only parental acceptance of their child’s asymmetry can determine treatment. Nevertheless, none of the cranial measurements used estimate esthetic severity, which is obviously subjective.
Radiological assessment of positional plagiocephaly is of little interest because simple cranial Xrays do not show the asymmetry. CT can be more useful to study cranial and basal asymmetry and also the asymmetry of the face, but the problem of irradiation for a growing patient limit its use to when true craniosynostosis is suspected [40, 41]. The need for anesthesia in patients older than 3 months is also a strong argument to forego radiological examination. Ultrasound has been used by some investigators, mainly to monitor sutures progression [42].
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In the last 10 years, infrared laser-assisted scansion of the skull in children has enabled monitoring of cranial modifications, providing an imprint of the skull at the beginning and of cranial development with or without treatment. Our experience confirmed the classical male predominance [16] of plagiocephaly, with 75% boys in our population. This could be explained by hormonal status in fetuses. Testosterone may accentuate muscular action in male fetuses, leading to torticollis, whereas relaxing hormones may loosen female connective tissues. [10, 22, 43] As already reported, multiple pregnancy favors plagiocephaly [10]. The large number of twins in our population (4.75%, versus 1% in the general population) supports this hypothesis (Figure 4).
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Antenatal ultrasound studies in a small cohort of pregnant women showed prenatal existence of cranial deformity in case of deformational plagiocephaly, suggesting that positional cranial deformation can already exist before birth.
At birth, the small size of the skull and the particular emotional atmosphere related to such
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an important event can explain the low rate of detection of cranial deformation. With growth, the larger skull volume reveals the deformation and eases diagnosis.
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In our experience, helmet therapy is an effective and well-tolerated treatment for plagiocephaly. Contrary to the opinion of some authors [32, 44], it seems particularly appropriate
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for correction of brachycephaly, reducing cranial index by 6.8% on average. In children with UDP, CDD was reduced by 0.78 cm (52%) on average. At end of treatment, in UDP, only 15.5% of
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children still showed severe plagiocephaly, requiring surgical treatment in a very few cases (0.2%). The helmet was also effective for facial asymmetry. The rate of facial symmetry increased from 4.2% at beginning of treatment to 62.4% at the end in brachycephaly, and from 2.5% to 40.2%, from 19.5% to 44.2% and from 17% to 77% in mild, moderate and severe forms of UDP, respectively. We were not able to constitute a control group, as our center adopted helmet treatment and developed the technique a long time ago, rapidly becoming a reference center for parents, who come specifically for that.
In a study comparing helmet therapy versus repositioning therapy, Graham et al. found that
helmet therapy led to a final CDD of 0.71 cm (61%), and was more effective than repositioning therapy. [10] In another non-randomized study, including 128 children with UDP, Kluba et al. also reported that cranial vault asymmetry index was reduced by 68% in children treated with a cranial helmet, versus only 31% in children without treatment. [17] Moreover, helmet therapy favored acquisition of facial symmetry in the present series, as also previously reported [16]. Like other authors [16], we think that helmet therapy does not impair global cranial growth, as the average
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cranial perimeter in our series was within 1 SD according to the WHO data. Consequently, our series confirms that helmet therapy optimizes and orients the orthopedic properties of the brain that are at the origin of skull growth. Efficacy depends on changing the pressure points in line with cranial modifications with skull growth and the ongoing correction obtained. Many other authors also consider that helmet therapy is effective [16–19, 22, 23, 27, 32, 35, 44–61]; even so, a few are skeptical [5, 26]. What is clear from our experience is that the helmet does not alter or impede skull growth. To demonstrate efficacy, serial CT-scans would be needed, to quantify skull progression and the changes in basal angles and cranial index; but this is unacceptable due to the high risk of cranial irradiation in young children.
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Changes in cerebrospinal fluid (CSF) dynamics are important in the development of positional plagiocephaly; according to some authors, increased amounts of CSF in subarachnoid spaces could trigger the skull deformation: i.e., external hydrocephaly could induce distortion of the skull. In true craniosynostosis, the involvement of CSF was reported by Carmel in 1981 [62] and, several
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years later, modifications of the subarachnoid spaces were described by Chadduck [63].
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Sawin, as well as other authors, demonstrated that the subarachnoid spaces of patients with occipital deformation were enlarged compared to controls [64–67].
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The subarachnoid space dilatation is similar to what is observed in idiopathic hydrocephalus, which is associated not with brain atrophy but with increased cranial circumference [64, 68].
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These modifications of subarachnoid spaces could be the primary anomaly that precedes skull deformation, or else the consequence of CSF circulation modifications induced by the modified cranial morphology. [64][69]. Dia et al. consequently suggested the simple term “occipital plagiocephaly”, because the two situations express the same problem and because the persistence of the external pressure could lead to true closure of the suture [8, 69]. These hypotheses give possible explanations for the flattening of the cranial deformation,
but neither explains the high incidence of positional plagiocephaly or the very rare incidence of lambdoid craniosynostosis [8].
The present rate of complications was very low. Side-effects were particularly rare (2.5%) and seldom serious. Moreover, in our experience, such minor complications disappear when treatment stops. In the present series, the incidence of such complications did not seem to be influenced by the type of plagiocephaly, although numbers were too small for comparative statistical analysis. Usually, the combination of plagiocephaly and brachycephaly, contrary to severity of deformation, is considered to correlate with a higher risk of complications [70]. 10
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Complication rate correlates with seasonal variation in temperature. During summertime, adherence to treatment needs more attention, with more frequent rest periods and more attentive skin care (drying and cleansing). For Freudlsperge et al., minor complications were far more frequent during helmet therapy (26.3%): pressure sores (13.7%), erythema (2.9%), skin erosion or infection (4.3%) or defective fitting (5.4%). [70] In some series, all parents declared at least one side-effect, with skin irritation frequency up to 96%.[5, 26] In these series, patients wore their helmet 23 hours a day, which is a little bit longer than recommended in our protocol. However, length of treatment has to be adapted to deformation progression, and generally, in our experience, to have a very good result, treatment duration is longer than that reported by some authors in the United States, which was
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generally 3 to 4 months. Finally, it is well recognized that helmet therapy is particularly safe and well-tolerated [18, 27] and does not negatively influence quality of life and of sleep [5]. In our experience, treatment was never interrupted for skin problems and there were no behavioral problems during follow-up until the age of 6 to 8 years.
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Our experience shows that poor parental compliance is a very important risk factor for helmet therapy failure [17] (RR = 2.42 [37]) and complications are almost always related to parental
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negligence. We are aware that simple and clear explanation of the goal and of the treatment protocol is essential to obtain good clinical results. Close follow-up at treatment outset is also mandatory to
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ensure a low rate of complications. [70] In our center, we are available to parents for any problems, as it is very important to provide an early answer and to react rapidly in case of complications or doubts. Simple explanations to parents who have to withdraw the device due to skin irritation avoid
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rapid worsening of the situation. The best attitude is to avoid the problem in the first place: prevention is essential.
Plagiocephaly prevention is an emerging concept. [5] Some authors suggest using
positioning aids from birth onward [22] and providing perinatal physiotherapy advice to parents to reduce cervical muscular contracture [16, 32, 50]. Measurement of cranial diameter should become an obligatory part of pediatric well-child consultations in the 3rd and 4th months of life. [22] Nutritional advice can be provided concerning the importance of adequate vitamin D intake during pregnancy and infancy. Vitamin D insufficiency during pregnancy is associated with skeletal homeostasis disorder in general and is independently associated with increased risk of skull deformation during infancy. Use of formula milk after birth is also associated with skull deformation [43]. Prevention of positional head deformities can involve physiotherapy [32, 55, 71, 72], osteopathy [50], counter-positioning [73], bedding devices [47] and cervical stretching [74]. Wilbrand et al. reported improvements in cranial shape in 50 children using non-orthotic methods 11
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(stretching exercises and adapted pillows). [32] However, pillows can cause side-effects (bilateral apostasis of the external ears). [32] A recent Australian study showed that, in a group of patients treated with helmet, correction of cranial parameters was better than in the group without helmet treatment. [75] Also, while there is no level 1 evidence of efficacy of helmet treatment, all the studies of anatomomorphological indices confirmed that helmets showed real morphological efficacy [23, 46, 48, 51, 54, 56, 60, 61]. While spontaneous reduction of UDP is reported in 70% cases [16, 18, 25, 48, 76, 77], in our experience severe cases never improved, in complete contradiction with the principle that, with the time and neuropsychological development, spontaneous recovery is always possible.
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After 30 years’ experience, we have learned that helmets can be effective up to the age of 19 months; it is very rare to observe significant improvement in skull shape after this age, and we stop treatment at that point. CONCLUSIONS
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Positional plagiocephaly and brachycephaly are a real problem in our society. The need for good quality of life and to avoid personal psychological problems has incited families and doctors
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to address this problem.
Our experience confirms that the cranial helmet is a simple and well-tolerated treatment
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which provides satisfactory clinical results in positional plagiocephaly. Success requires good teamwork with parents and careful management by orthotist, physiotherapist and physician to avoid
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minor side-effects. Helmet therapy must be applied as early as possible, to take full advantage of cranial bone plasticity. Unfortunately, this alternative treatment remains relatively under-used in France, undoubtedly because few pediatric neurosurgical departments propose it. Nevertheless, there remain many controversies, in particular concerning long-term esthetic and functional outcome. A long-term multicenter prospective study is needed to remove doubts. Finally, “acceptable” head shape in infants and at an older age is subjective. What is certain
is that cranial asymmetry persists into adolescence and adulthood. The psychological consequences and acceptance by parents and child are less certain. As Montaigne said, "It is better to have a well-structured head than a full head" - a wellstructured head with a nice craniofacial appearance! [78].
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FIGURE LEGENDS Figure 1: Diagram of the force applied by the helmet to remodel the head shape. Figure 2 A and B: Pictures of children wearing a helmet Figure 3: 3D measurement for tailored helmet.
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Figure 4: Picture of twins wearing a helmet.
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23. Lee RP, Teichgraeber JF, Baumgartner JE, et al. (2008) Long-term treatment effectiveness of molding helmet therapy in the correction of posterior deformational plagiocephaly: a five-year followup. Cleft Palate-Craniofacial J 45:240–245. https://doi.org/10.1597/06-210.1 24. Steinbok P, Lam D, Singh S, et al. (2007) Long-term outcome of infants with positional occipital plagiocephaly. Childs Nerv Syst 23:1275–1283. https://doi.org/10.1007/s00381-007-0373-y
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25. Collett BR, Gray KE, Starr JR, et al. (2013) Development at age 36 months in children with deformational plagiocephaly. Pediatrics 131:e109-115. https://doi.org/10.1542/peds.2012-1779
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Kluba S, Kraut W, Reinert S, Krimmel M (2011) What is the optimal time to start helmet
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therapy in positional plagiocephaly? Plast Reconstr Surg 128:492–498. https://doi.org/10.1097/PRS.0b013e31821b62d6 36. Thompson JT, David LR, Wood B, et al. (2009) Outcome analysis of helmet therapy for positional plagiocephaly using a three-dimensional surface scanning laser. J Craniofac Surg 20:362– 365. https://doi.org/10.1097/SCS.0b013e3181992382 37. Steinberg JP, Rawlani R, Humphries LS, et al. (2015) Effectiveness of conservative therapy and helmet therapy for positional cranial deformation. Plast Reconstr Surg 135:833–842. https://doi.org/10.1097/PRS.0000000000000955 38. Di Rocco F, Ble V, Beuriat P-A, et al. (2019) Prevalence and severity of positional plagiocephaly in children and adolescents. Acta Neurochir (Wien) 161:1095–1098. https://doi.org/10.1007/s00701-019-03924-2
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39. Argenta L, David L, Thompson J (2004) Clinical classification of positional plagiocephaly. J Craniofac Surg 15:368–372 40. Brenner D, Elliston C, Hall E, Berdon W (2001) Estimated risks of radiation-induced fatal cancer from pediatric CT. AJR Am J Roentgenol 176:289–296. https://doi.org/10.2214/ajr.176.2.1760289
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41. Pearce MS, Salotti JA, Little MP, et al. (2012) Radiation exposure from CT scans in childhood and subsequent risk of leukaemia and brain tumours: a retrospective cohort study. Lancet Lond Engl 380:499–505. https://doi.org/10.1016/S0140-6736(12)60815-0
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42. Sze RW, Parisi MT, Sidhu M, et al. (2003) Ultrasound screening of the lambdoid suture in the child with posterior plagiocephaly. Pediatr Radiol 33:630–636. https://doi.org/10.1007/s00247-0031009-3 43. Weernink MGM, van Wijk RM, Groothuis-Oudshoorn CGM, et al. (2016) Insufficient vitamin D supplement use during pregnancy and early childhood: a risk factor for positional skull deformation. Matern Child Nutr 12:177–188. https://doi.org/10.1111/mcn.12153
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44. Teichgraeber JF, Seymour-Dempsey K, Baumgartner JE, et al. (2004) Molding helmet therapy in the treatment of brachycephaly and plagiocephaly. J Craniofac Surg 15:118–123 45. Bialocerkowski AE, Vladusic SL, Howell SM (2005) Conservative interventions for positional plagiocephaly: a systematic review. Dev Med Child Neurol 47:563–570 46. Couture DE, Crantford JC, Somasundaram A, et al. (2013) Efficacy of passive helmet therapy for deformational plagiocephaly: report of 1050 cases. Neurosurg Focus 35:E4. https://doi.org/10.3171/2013.8.FOCUS13258 47. de Chalain T (2003) The Safe-T-Sleep device: safety and efficacy in maintaining infant sleeping position. N Z Med J 116:U581 48. Goh JL, Bauer DF, Durham SR, Stotland MA (2013) Orthotic (helmet) therapy in the treatment of plagiocephaly. Neurosurg Focus 35:E2. https://doi.org/10.3171/2013.7.FOCUS13260 49. Govaert B, Michels A, Colla C, van der Hulst R (2008) Molding therapy of positional plagiocephaly: subjective outcome and quality of life. J Craniofac Surg 19:56–58. https://doi.org/10.1097/SCS.0b013e31815c8a27 50. Lessard S, Gagnon I, Trottier N (2011) Exploring the impact of osteopathic treatment on cranial asymmetries associated with nonsynostotic plagiocephaly in infants. Complement Ther Clin Pract 17:193–198. https://doi.org/10.1016/j.ctcp.2011.02.001
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51. Lipira AB, Gordon S, Darvann TA, et al. (2010) Helmet versus active repositioning for plagiocephaly: a three-dimensional analysis. Pediatrics 126:e936-945. https://doi.org/10.1542/peds.2009-1249 52. Mortenson P, Steinbok P, Smith D (2012) Deformational plagiocephaly and orthotic treatment: indications and limitations. Childs Nerv Syst 28:1407–1412. https://doi.org/10.1007/s00381-012-1755-3 53. Mulliken JB, Vander Woude DL, Hansen M, et al. (1999) Analysis of posterior plagiocephaly: deformational versus synostotic. Plast Reconstr Surg 103:371–380 54. Naidoo SD, Skolnick GB, Patel KB, et al. (2015) Long-term outcomes in treatment of deformational plagiocephaly and brachycephaly using helmet therapy and repositioning: a longitudinal cohort study. Childs Nerv Syst 31:1547–1552. https://doi.org/10.1007/s00381-015-2769-4
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55. Neufeld S, Birkett S (2000) What to do about flat heads: preventing and treating positional occipital flattening. Axone Dartm NS 22:29–31 56. Plank LH, Giavedoni B, Lombardo JR, et al. (2006) Comparison of infant head shape changes in deformational plagiocephaly following treatment with a cranial remolding orthosis using a noninvasive laser shape digitizer. J Craniofac Surg 17:1084–1091. https://doi.org/10.1097/01.scs.0000244920.07383.85
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57. Robinson S, Proctor M (2009) Diagnosis and management of deformational plagiocephaly. J Neurosurg Pediatr 3:284–295. https://doi.org/10.3171/2009.1.PEDS08330
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