Effectiveness of pediatric integrative manual therapy in cervical movement limitation in infants with positional plagiocephaly: a randomized controlled trial.

BACKGROUND: Positional plagiocephaly (PP) is a cranial deformation frequent amongst children and consisting in a flattened and asymmetrical head shape. PP is associated with excessive time in supine and with congenital muscular torticollis (CMT). Few studies have evaluated the efficiency of a manual therapy approach in PP. The purpose of this parallel randomized controlled trial is to compare the effectiveness of adding a manual therapy approach to a caregiver education program focusing on active rotation range of motion (AROM) and neuromotor development in a PP pediatric sample. METHODS: Thirty-four children with PP and less than 28 week-old were randomly distributed into two groups. AROM and neuromotor development with Alberta Infant Motor Scale (AIMS) were measured. The evaluation was performed by an examiner, blinded to the randomization of the subjects. A pediatric integrative manual therapy (PIMT) group received 10-sessions involving manual therapy and a caregiver education program. Manual therapy was addressed to the upper cervical spine to mobilize the occiput, atlas and axis. The caregiver educational program consisted in exercises to reduce the positional preference and to stimulate motor development. The control group received the caregiver education program exclusively. To compare intervention effectiveness across the groups, improvement indexes of AROM and AIMS were calculated using the difference of the final measurement values minus the baseline measurement values. If the distribution was normal, the improvement indexes were compared using the Student t-test for independent samples; if not, the Mann-Whitney U test was used. The effect size of the interventions was calculated using Cohen's d. RESULTS: All randomized subjects were analysed. After the intervention, the PIMT group showed a significantly higher increase in rotation (29.68 ± 18.41°) than the control group (6.13 ± 17.69°) (p = 0.001). Both groups improved the neuromotor development but no statistically significant differences were found. No harm was reported during the study. CONCLUSION: The PIMT intervention program was more effective in increasing AROM than using only a caregiver education program. The study has been retrospectively registered at clinicaltrials.gov, with identification number NCT03659032 . Registration date: September 1, 2018.

Do Infant Motor Skills Mediate the Association Between Positional Plagiocephaly/Brachycephaly and Cognition in School-Aged Children?

OBJECTIVE: Positional plagiocephaly/brachycephaly (PPB) is associated with lower cognitive scores in school-aged children. This study tested the hypothesis that infant motor skills mediate this association. METHODS: Children with a history of PPB (cases, n = 187) and without PPB (controls, n = 149) were followed from infancy through approximately 9 years of age. Infant motor skills were assessed using the Bayley Scales of Infant and Toddler Development, 3rd edition (Bayley-3), and cognition was assessed using the Differential Ability Scales, 2nd edition (DAS-2). The Bayley-3 motor composite was examined as a mediator of the association between PPB and DAS-2 general cognitive ability (GCA) scores. In secondary analyses, mediation models were examined for the DAS-2 verbal ability, nonverbal ability, and working memory scores; models using the Bayley-3 fine versus gross motor scores also were examined. RESULTS: Cases scored lower than controls on the DAS-GCA (ß = -4.6; 95% CI = -7.2 to -2.0), with an indirect (mediated) effect of ß = -1.5 (95% CI = -2.6 to -0.4) and direct effect of ß = -3.1 (95% CI = -5.7 to -0.5). Infant motor skills accounted for approximately 33% of the case-control difference in DAS-2 GCA scores. Results were similar for other DAS-2 outcomes. Evidence of mediation was greater for Bayley-3 gross motor versus fine motor scores. CONCLUSION: Infant motor skills partially mediate the association between PPB and cognition in school-aged children. Monitoring motor development and providing intervention as needed may help offset associated developmental concerns for children with PPB. IMPACT: To our knowledge, this study is the first longitudinal investigation of the development of children with and without PPB from infancy through the early school years and the first to examine motor skills as a mediator of cognitive outcomes in this population. The findings highlight the importance of early motor skills for other developmental outcomes. LAY SUMMARY: Infants' motor skills are related to the development of PPB and its association with later cognition. If your child has PPB, physical therapists may have an important role in assessing and providing treatment to promote motor development.

Motor Function in School-Aged Children With Positional Plagiocephaly or Brachycephaly.

OBJECTIVE: To determine whether children with a history of positional plagiocephaly/brachycephaly (PPB) show persistent deficits in motor development. METHODS: In a longitudinal cohort study, we completed follow-up assessments with 187 school-aged children with PPB and 149 participants without PPB who were originally enrolled in infancy. Primary outcomes were the Bruininks-Oseretsky Test of Motor Proficiency-Second Edition (BOT-2) composite scores. RESULTS: Children with PPB scored lower than controls on the BOT-2. Stratified analyses indicated that differences were restricted to children who had moderate-severe PPB. No consistent differences were observed in children who had mild PPB. CONCLUSION: Children who had moderate-severe PPB in infancy show persistent differences in motor function. We suggest close developmental monitoring and early intervention to address motor deficits.

Infant/toddler motor skills as predictors of cognition and language in children with and without positional skull deformation.

PURPOSE: To estimate associations between early motor abilities (at two age points, 7 and 18 months on average) and cognitive/language outcomes at age 3. To determine whether these associations are similar for children with and without positional plagiocephaly and/or brachycephaly (PPB). METHODS: The Bayley Scales of Infant/Toddler Development 3 were given at all age points to 235 children with PPB and 167 without PPB. Linear regressions assessed longitudinal associations between fine and gross motor scales and cognition/language. Item analyses examined the contributions of specific motor skills. RESULTS: Associations between 7-month motor skills and cognition/language were modest overall (effect sizes [ES] = - 0.08 to 0.10, p = .13 to .95). At 18 months, both fine and gross motor skills were associated with outcomes for children with PPB (ES = 0.21 to 0.41, p < .001 to .01), but among those without PPB, only fine motor skills were associated with outcomes (ES = 0.21 to 0.27, p < .001 to .001). CONCLUSIONS: Toddlers' motor skills were associated with cognition and language at 3 years, particularly among children with PPB. Interventions targeting early motor development in infants and toddlers with PPB may have downstream benefits for other outcomes.

Six month-old infants with deformational plagiocephaly do not differ from unaffected infants with respect to vocal control.

OBJECTIVES: The recommendation of a supine sleeping position led to a significant reduction of SIDS, but increased positional skull deformities (DP). Here, a quantitative analysis of babbling aims to complement previous studies of language-relevant competence based on items of the Bayley-scales that suggested the presence of developmental language delays in DP infants. Measures of fundamental frequency variability as proxies for vocal control are well suited for testing this assumption, since the laryngeal neuro-muscular system matures early and is coupled with brain function while working rapidly in coordinating the structures and mechanisms involved in infant sound production. METHODS: Sixty-six healthy, full-term infants with normal hearing and a monolingual background took part: (1) moderately asymmetrical DP group - N = 41; 21 male; (2) severely asymmetrical DP group - N = 10; 8 male; and (3) controls - N = 15; 5 male (group assignment based on stereophotogrammetric 360° scans). Fundamental frequency (fo) measures were taken as proxies for vocal control skills during babbling. RESULTS: A MANOVA revealed no significant multivariate effect for the shape group, Wilks' λ = 0.86, F(2, 63) = 1.21, p = 0.30, η2 = 0.07 (medium effect-size). The results do not support previous findings based on Bayley scale evaluations that suggested a negative impact of DP on language development during infancy. CONCLUSIONS: A strong link between DP and brain dysfunction affecting vocal control, which would cause deviations in otherwise healthy DP infants, was not observed. Objective long-term studies of sound production are necessary to identify and/or understand the potential consequences of DP on early language development.

[Effects of deformational plagiocephaly during the first 12 months on the psychomotor development of prematurely born infants]. / Effets de la plagiocéphalie posturale au cours des 12 premiers mois sur le développement psychomoteur à 4 ans des enfants nés très prématurément.

AIMS: The link between deformational plagiocephaly and psychomotor development is a recurrent question in medical publications. Main publications concentrate on term infants, but there is a lack of data on the impact of deformational plagiocephaly on the long-term neurodevelopment of premature infants. We attempted to establish a possible relation between deformational plagiocephaly during the 1st year of life and the psychomotor score at 4 years in prematurely born infants. Other risk factors potentially impacting the psychomotor score were also studied. MATERIAL AND METHODS: A retrospective study of the files of the children followed by the "Naître et Devenir Région PACA Ouest Corse Sud" healthcare network and included in the database allowed us to select a cohort of 594 infants born prematurely at under 33 weeks of gestational age. These children were developmentally evaluated during the 1st year of life and at 4 years or age using the "EVAL Mater" test. The "Naître et Devenir" network is following up infants born prematurely at under 33 weeks of gestation in the West Provence Alpes Côte d'Azur and South Corsica region, from discharge to 7 years. A group of 170 specially trained pediatricians follow these infants developmentally at term, 3, 6, 9, 12, 18, and 24 months of corrected age and 3, 4 5, 6, and 7 years. Data are collected in a specially designed database. RESULTS: There was no significant link between deformational plagiocephaly during the 1st year of life and a pathological psychomotor score at age 4, but some risk factors were demonstrated: male gender, birth at under 28 weeks of gestational age, weight at birth under 1000g, having a Latal and Ferriero neuromotor score equal to or greater than 2 at 3 months of corrected age, and to a lesser extent having a prescription for physiotherapy during the 1st year. CONCLUSION: The research on deformational plagiocephaly in the full-term infant suggests a relation between deformational plagiocephaly and developmental delay predominantly on the motor side, with an increased rate of special needs services at school age. The question is raised of whether deformational plagiocephaly is the cause of the delay or an early sign of cerebral anomaly with an early motor delay in full-term infants. The results suggest that deformational plagiocephaly in the prematurely born infant may not be related to neurodevelopmental delay but simply to the extended time spent in the supine position because of the early birth associated with physiological hypotonia and axial extension. Other risk factors such as male gender, birth before 28 weeks of gestation, weight less than 1000g, a Latal and Ferriero neuromotor score greater than 2 at 3 months of corrected age, and having a prescription for physiotherapy during the 1st year of life are strongly related to delayed psychomotor development at age 4.

Deformational plagiocephaly in normal infants: a systematic review of causes and hypotheses.

BACKGROUND: Deformational plagiocephaly (DP) is one of the most prevalent abnormal findings in infants and a frequent reason for parents to seek paediatric advice. OBJECTIVE: To systematically review the literature and identify evidence and hypotheses on the aetiology and determinants of DP in otherwise healthy infants. DESIGN: Systematic keyword search in all major biomedical databases to identify peer-reviewed publications reporting (a) empirical research or (b) hypotheses on the aetiology of DP in healthy, term infants. 3150 studies published between 1985 and 2016 and containing relevant keywords were screened. In a two-pronged approach, results were summarised separately for the body of empirical work (22 studies) and the body of hypotheses (110 articles). REVIEW FINDINGS: Only a few empirical studies have examined risk factors in non-selected patient populations on a higher grade methodological level. The most commonly reported risk factors were: male gender, supine sleep position, limited neck rotation or preference in head position, first-born child, lower level of activity and lack of tummy time. Agreement between empirical studies was poor for most exposures, including supine sleep position, tummy time and use of car seats. The articles reporting hypotheses on the aetiology of DP cover a wide field of environmental and biological factors, but include little suggestions as to the potential influence of the everyday care environment of the baby. CONCLUSIONS AND RELEVANCE: The evidence on the aetiology of DP is fragmentary and heterogeneous. In addition, factors possibly relevant to the development of DP have not been appreciated in the scientific discussion.

Assessing the Cervical Range of Motion in Infants With Positional Plagiocephaly.

PURPOSE: To determine if infants with positional plagiocephaly have limitations of active and passive cervical range of motion measured with simple and reliable methods. METHODS: The examiners assessed bilateral active and passive cervical rotations and passive cervical lateral flexion. Cervical assessment was performed twice by 2 different physicians to assess intertester reliability. To assess intratester reliability the first investigator performed a second examination 48 hours after the first one. RESULTS: One-hundred nine subjects were analyzed; 70.7% of the sample had head positional preference on the right, while 29.3% had head positional preference on the left (χ 35.52, P <0.001). Cervical rotations and lateral flexion showed reliable levels of agreement for intra and intertester reliability. CONCLUSIONS: The most limited range of motion in infants with positional plagiocephaly was cervical active rotation which affected more than 90% of patients. Passive cervical rotations and lateral flexion were limited in more than 60% of patients.

Preventing deformational plagiocephaly through parent guidance: a randomized, controlled trial.

Deformational plagiocephaly (DP) occurs frequently in otherwise healthy infants. Many infants with DP undergo physiotherapy or helmet therapy, and ample treatment-related research is available. However, the possibility of preventing DP has been left with little attention. We sought to evaluate the effectiveness of intervention in the newborn's environment, positioning, and handling on the prevalence of DP at 3 months and to investigate the causal relationship between DP and cervical imbalance. We carried out a randomized controlled trial, with healthy newborns randomized into two groups at birth. All families received standard positioning instructions to prevent SIDS. Additionally, the intervention group received detailed instructions regarding the infant's environment, positioning, and handling, with the goal of creating a nonrestrictive environment that promotes spontaneous physical movement and symmetrical motor development. Two- and three-dimensional photogrammetry served to assess cranial shape and goniometry to measure cervical motion. At 3 months, the prevalence of DP was lower in the intervention group in both 2D (11 vs 31 %) and 3D analyses (15 vs 33 %), and the asymmetry was milder in the intervention group. Infants with DP at follow-up had also developed more torticollis. CONCLUSION: An early educational intervention reduces the prevalence and severity of DP at 3 months. WHAT IS KNOWN: •Deformational plagiocephaly, often with associated torticollis, is common in healthy infants. •Parental education is frequently recommended for preventing deformational plagiocephaly, although information regarding the effectiveness of preventive strategies is scarce. WHAT IS NEW: •Early parent guidance effectively reduces the prevalence and severity of DP and improves the cervical range of motion at three months. •Educating both parents and professionals about proper infant positioning on a national scale could help minimize public healthcare costs.

Brain electrophysiology reveals intact processing of speech sounds in deformational plagiocephaly.

BACKGROUND: The prevalence of deformational plagiocephaly has risen dramatically in recent years, now affecting 15 percent or more of infants. Prior research using developmental scales suggests that these children may be at elevated risk for developmental delays. However, the low positive predictive value of such instruments in identifying long-term impairment, coupled with their poor reliability in infants, warrants the development of methods to more precisely measure brain function in craniofacial patients. Event-related potentials offer a direct measure of cortical activity that is highly applicable to young populations and has been implemented in other disorders to predict long-term cognitive functioning. The current study used event-related potentials to contrast neural correlates of auditory perception in infants with deformational plagiocephaly and typically developing children. METHODS: Event-related potentials were recorded while 16 infants with deformational plagiocephaly and 18 nonaffected controls passively listened to speech sounds. Given prior research suggesting their association with subsequent functioning, analyses focused on the P150 and N450 event-related potential components. RESULTS: Deformational plagiocephaly patients and normal controls showed comparable cortical responses to speech sounds at both auditory event-related potential components. CONCLUSIONS: Children with deformational plagiocephaly demonstrate neural responses to language that are consistent with normative expectations and comparable to those of typical children. These results indicate that head shape deformity secondary to supine sleep is not associated with impairments in auditory processing. The applicability of the current methods in early infancy suggests that electrophysiologic brain recordings represent a promising method of monitoring brain development in children with cranial disorders. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, II.

Congenital muscular torticollis and positional plagiocephaly.

On the basis of observational studies, child health practitioners in primary care settings should consider the diagnosis of congenital muscular torticollis (CMT)in infants with risk factors from birth history for intrauterine malpositioning or constraint (C). On the basis of observational studies, CMT is often associated with other conditions, including positional plagiocephaly and gross motor delays from weakened truncal muscles and/or lack of head control in early infancy (C). On the basis of observational studies, child health practitioners should counsel parents that infants should be on their stomachs frequently whenever they are awake and under direct adult supervision to develop their prone motor skills (C). On the basis of consensus, early identification of CMT(with or without positional plagiocephaly) and prompt referral to a physical therapist experienced in the treatment of CMT should be considered to avoid more costly or invasive treatments, such as cranial orthoses or surgery (D).

Early neurodevelopment in infants with deformational plagiocephaly.

The objective of this study was to characterize the early neurodevelopmental profile of Australian infants with deformational plagiocephaly (DP). Twenty-one infants with a confirmed diagnosis of DP (mean age, 7.9 months; SD, 2.0 months) were assessed on the Bayley Scales of Infant Development, Second Edition, before treatment. As a group, infants with DP showed significantly weaker motor skills compared with the normative population. Patients with DP did not significantly differ from normative population estimates with respect to cognitive abilities. Males with DP displayed significantly reduced motor abilities compared to females. These findings do not suggest that DP causes developmental problems, but DP could be a marker for risk of developmental problems. Screening for developmental concerns in infants with DP is therefore important.

Clinical course of asymmetric motor performance and deformational plagiocephaly in very preterm infants.

OBJECTIVES: To describe the clinical courses of positional preference and deformational plagiocephaly up to 6 months corrected age (CA) in infants born at gestational age <30 weeks or birth weight <1000 g, and to explore predictive factors for the persistence of these phenomena. STUDY DESIGN: A total of 120 infants were examined 3 times each. The presence of deformational plagiocephaly and a score of 0-6 on an asymmetry performance scale served as outcome measures at 6 months CA. Predictive factors were determined using regression analysis. RESULTS: The prevalence of a positional preference of the head was 65.8% (79 of 120) at term-equivalent age (TEA) and 36.7% (44 of 120) at 3 months CA and that of deformational plagiocephaly was 30% (36 of 120) at TEA and 50% (60 of 120) at 3 months CA. At 6 months CA, 15.8% of the infants (19 of 120) scored ≥ 2 of a possible 6 on the asymmetry performance scale and 23.3% (28 of 120) had deformational plagiocephaly. Sleeping in the supine position was predictive of an asymmetric motor performance at 6 months CA. Chronic lung disease and/or slow gross motor maturation at 3 months CA predicted the persistence of deformational plagiocephaly. CONCLUSION: Infants born very preterm may develop deformational plagiocephaly. A positional preference of the head at TEA seems to be a normal aspect of these infants' motor repertoire, with limited ability to predict persistence of an asymmetric motor performance. The decreased prevalence of deformational plagiocephaly between 3 and 6 months CA indicates an optimistic course. Infants with a history of chronic lung disease and/or slow gross motor maturation merit timely intervention.

Development at age 36 months in children with deformational plagiocephaly.

OBJECTIVES: Infants and toddlers with deformational plagiocephaly (DP) have been shown to score lower on developmental measures than unaffected children. To determine whether these differences persist, we examined development in 36-month-old children with and without a history of DP. METHODS: Participants included 224 children with DP and 231 children without diagnosed DP, all of who had been followed in a longitudinal study since infancy. To confirm the presence or absence of DP, pediatricians blinded to children's case status rated 3-dimensional cranial images taken when children were 7 months old on average. The Bayley Scales of Infant and Toddler Development, Third Edition (BSID-III) was administered as a measure of child development. RESULTS: Children with DP scored lower on all scales of the BSID-III than children without DP. Differences were largest in cognition, language, and parent-reported adaptive behavior (adjusted differences = -2.9 to -4.4 standard score points) and smallest in motor development (adjusted difference = -2.7). Children in the control group who did not have previously diagnosed DP but who were later rated by pediatricians to have at least mild cranial deformation also scored lower on the BSID-III than unaffected controls. CONCLUSIONS: Preschool-aged children with a history of DP continue to receive lower developmental scores than unaffected controls. These findings do not imply that DP causes developmental problems, but DP may nonetheless serve as a marker of developmental risk. We encourage clinicians to screen children with DP for developmental concerns to facilitate early identification and intervention.

Cosmetic and cognitive outcomes of positional plagiocephaly treatment.

PURPOSE: Positional plagiocephaly is an acquired deformation of an intrinsically normal infant skull by sustained or excessive extrinsic forces. Non-surgical techniques include counter-positioning, supervised prone time and orthotic molding for more refractory cases. Long-term effects of positional plagiocephaly on development remain undefined, and this study evaluated cosmetic and cognitive outcomes of plagiocephaly management. METHOD: Surveys were administered to parents of patients treated for positional plagiocephaly through the Children's Hospital of Eastern Ontario. Categorical responses interrogated cosmetic outcome, school performance, language skills, cognitive development and societal function. Pearson coefficient analysis tested outcomes dependency on gender, age, and plagiocephaly side at the 0.05 level of significance. RESULTS: Eighty respondents (51 male, 29 female) were divided as 58 right- and 22 left-sided pathology. Positional therapy was uniformly applied, and a helmet orthosis was utilized in 36% of cases. Median follow-up age was nine years with normal head appearance in 75% of cases. Only 4% of parents and 9% of patients observed significant residual asymmetry. These results did not vary by gender, age or deformity side. Left-sided disease predicted poorer language development and academic performance. Expressive speech abnormality occurred in twice as many patients with left-sided disease (36% versus 16%, p=0.04) along with three-fold greater special education requirements (27% versus 10%, p=0.04). CONCLUSIONS: Non-surgical plagiocephaly management achieved good cosmetic outcome among patients in this study. Children with left-sided disease frequently encountered difficulties with cognitive and scholastic endeavors, although the roles of the underlying disease and the treatment measures in this delay cannot be differentiated.

Does helmet therapy influence the ear shift in positional plagiocephaly?

BACKGROUND: Helmet therapy is widely accepted in the treatment of severe positional plagiocephaly. The improvement of the cranial asymmetry under therapy is evident, but parents are also concerned about the ear shift. Our study investigated the influence of helmet therapy on the position of the ears and analyzed the reliability of clinical observations regarding cranial asymmetry and ear shift. METHODS: Three-dimensional stereophotogrammetry of 80 infants with severe positional plagiocephaly was performed before and after helmet therapy. The cranial vault asymmetry index (CVAI) and ear shift were measured and statistically compared. The correlation between the change of CVAI and ear shift was investigated. Three surgeons visually evaluated the treatment results on three-dimensional images independently with a standard questionnaire. The results were compared with the three-dimensional measurements. RESULTS: Sixty infants had a relevant initial ear shift. Under therapy, the shift was improved significantly by a mean of 29.8% (P < 0.001). Twenty infants with an initial straight ear line statistically deteriorate under therapy (P < 0.0001). We found no strong linear correlation between the changes of the CVAI and the ear shift. Analysis of the questionnaire revealed a good correlation between the clinical impression and three-dimensional measurements for the head asymmetry, whereas observations regarding changes in the ear shift were not reliable. CONCLUSIONS: Helmet treatment significantly improves an initial malposition of the external ear in infants with positional plagiocephaly. A severe ear shift can be associated with a moderate CVAI and vice versa. In contrast to the CVAI, small changes of the ear shift cannot be evaluated reliably by clinical investigation.

HElmet therapy Assessment in infants with Deformed Skulls (HEADS): protocol for a randomised controlled trial.

BACKGROUND: In The Netherlands, helmet therapy is a commonly used treatment in infants with skull deformation (deformational plagiocephaly or deformational brachycephaly). However, evidence of the effectiveness of this treatment remains lacking. The HEADS study (HElmet therapy Assessment in Deformed Skulls) aims to determine the effects and costs of helmet therapy compared to no helmet therapy in infants with moderate to severe skull deformation. METHODS/DESIGN: Pragmatic randomised controlled trial (RCT) nested in a cohort study. The cohort study included infants with a positional preference and/or skull deformation at two to four months (first assessment). At 5 months of age, all children were assessed again and infants meeting the criteria for helmet therapy were asked to participate in the RCT. Participants were randomly allocated to either helmet therapy or no helmet therapy. Parents of eligible infants that do not agree with enrolment in the RCT were invited to stay enrolled for follow up in a non-randomisedrandomised controlled trial (nRCT); they were then free to make the decision to start helmet therapy or not. Follow-up assessments took place at 8, 12 and 24 months of age. The main outcome will be head shape at 24 months that is measured using plagiocephalometry. Secondary outcomes will be satisfaction of parents and professionals with the appearance of the child, parental concerns about the future, anxiety level and satisfaction with the treatment, motor development and quality of life of the infant. Finally, compliance and costs will also be determined. DISCUSSION: HEADS will be the first study presenting data from an RCT on the effectiveness of helmet therapy. Outcomes will be important for affected children and their parents, health care professionals and future treatment policies. Our findings are likely to influence the reimbursement policies of health insurance companies.Besides these health outcomes, we will be able to address several methodological questions, e.g. do participants in an RCT represent the eligible target population and do outcomes of the RCT differ from outcomes found in the nRCT? TRIAL REGISTRATION: ISRCTN18473161.

Serial developmental assessments in infants with deformational plagiocephaly.

AIM: An association between positional plagiocephaly and developmental problems has previously been noted, but whether delays persist over time has not been established. This study aimed to determine developmental outcomes for children with deformational plagiocephaly over 1 year of follow up. METHODS: This was a longitudinal cohort study of 126 infants with deformational plagiocephaly recruited at an outpatient clinic. Development was assessed with the parent-completed Ages and Stages Questionnaires at recruitment and repeated at follow-up assessments in the home 3, 6 and 12 months later. Questionnaires were scored according to cut-off scores from the Ages and Stages Questionnaires, Third Edition. RESULTS: Ninety-six percent of children were followed up for the full 12 months. The existence of one or more delays initially was 30%; this rose to 42% at the 3-month follow up then dropped back to 23% by the 12-month follow up. Delays were predominantly in the gross motor domain. Ten percent had > 4 delays in total over the four assessments. Mothers with tertiary education were more likely to have infants showing delays that persisted over time. CONCLUSIONS: Infants with deformational plagiocephaly exhibited marked delays especially in early infancy. These delays were largely gross motor in type but had reduced to approach the expected level by the time of the 12-month follow up, at a mean age of 17 months.

Classification and pathogenic models of unintentional postural cranial deformities in infants: plagiocephalies and brachycephalies.

Unintentional postural deformities of the skull have increased in a pseudoepidemic manner in the last 15 years. Although dorsal decubitus and prenatal risk factors can play a role in the genesis of such deformities, we think that a crucial determinant is a postnatal defect of cervical mobility responsible for the infant's posture (ie, positional preference) when supine. Indeed, muscular factors, which limit the range of head and neck movements, have been underestimated in the genesis of skull deformities. Here, we have retrospectively analyzed data from 181 infants with unintentional skull deformities and propose a classification of these deformities into 3 types based on their pathogenic model and clinical appearance: fronto-occipital plagiocephalies due to severe muscle hypertonia in which the myogenic component is the first implicated, occipital plagiocephalies with muscle imbalance due to neurogenic muscle hypertonia, and posterior brachycephalies with neurogenic muscle hypertonia of the suboccipital muscles due to trauma to the occipitovertebral junction. Future studies on the size and density of specific muscles or group of muscles should help us to better understand their involvement in the pathogenesis of postural deformities. Our findings also highlight the importance of carefully assessing cervical mobility during the first week of life to detect possible limitations and to prescribe (if needed) an adapted rehabilitation. Rehabilitation should be associated with postural measures put in place when infants sleep supine to prevent the appearance of skull deformations.

Case-control study of neurodevelopment in deformational plagiocephaly.

OBJECTIVE: We assessed the neurodevelopment of infants with and without deformational plagiocephaly (DP), at an average age of 6 months. METHODS: The Bayley Scales of Infant Development III (BSID-III) were administered to 235 case subjects and 237 demographically similar, control participants. Three-dimensional head photographs were randomized and rated for severity of deformation by 2 craniofacial dysmorphologists who were blinded to case status. RESULTS: We excluded 2 case subjects with no photographic evidence of DP and 70 control subjects who were judged to have some degree of DP. With control for age, gender, and socioeconomic status, case subjects performed worse than control subjects on all BSID-III scales and subscales. Case subjects' average scores on the motor composite scale were approximately 10 points lower than control subjects' average scores (P < .001). Differences for the cognitive and language composite scales were approximately 5 points, on average (P < .001 for both scales). In subscale analyses, case subjects' gross-motor deficits were greater than their fine-motor deficits. Among case subjects, there was no association between BSID-III performance and the presence of torticollis or infant age at diagnosis. CONCLUSIONS: DP seems to be associated with early neurodevelopmental disadvantage, which is most evident in motor functions. After follow-up evaluations of this cohort at 18 and 36 months, we will assess the stability of this finding. These data do not necessarily imply that DP causes neurodevelopmental delay; they indicate only that DP is a marker of elevated risk for delays. Pediatricians should monitor closely the development of infants with this condition.