Comorbidities of deformational plagiocephaly in infancy: A scoping review.

AIM: While deformational plagiocephaly (DP) is suspected to be associated with comorbidities, their nature and prevalence are unclear. This scoping review aims to report DP comorbidities occurring until the age of 2 years, their prevalence and whether they depend on the child's age and sex. METHODS: Relevant studies were identified by searching the Cochrane, MEDLINE, EMBASE, PubMed and EBSCO databases from 1992 to 30 April 2021. Data on study characteristics, comorbidities and assessment instruments were extracted and qualitatively synthesised. Risk of bias was assessed and studies with high risk of bias were excluded. RESULTS: Studies meeting selection criteria (n = 27) often evaluated groups from tertiary clinics, implying selection bias. Studies reported on developmental delay (n = 16), limited speech production (n = 1), auditory (n = 3), visual (n = 3), mandibular (n = 3) and neurological impairments (n = 1). The data did not allow prevalence calculation or modifying effect of sex. Due to biased data, the review provided no evidence on DP comorbidities. Weak evidence suggested that in the selective samples, DP was associated with motor and language delays in the first year. CONCLUSION: Due to biased data, no evidence on comorbidity in infants with DP was available. Our study underlined the need of risk of bias assessment in scoping reviews.

Neurocognitive Outcomes in Deformational Plagiocephaly: Is There an Association between Morphologic Severity and Results?

BACKGROUND: The neurodevelopmental effects of skull asymmetry and orthotic helmet therapy for deformational plagiocephaly (DP) have had limited investigation. This study assessed the long-term neurocognitive outcomes in patients with DP and their association with orthotic helmet therapy and head shape abnormality. METHODS: A total of 138 school-age children with a history of DP, 108 of whom received helmet therapy, were tested with a neurocognitive battery assessing academic achievement, intelligence quotient, and visual-motor function. Severity of presenting plagiocephaly was calculated using anthropometric and photometric measurements. Analysis of covariance was used to compare outcomes between helmeted and nonhelmeted cohorts, unilateral plagiocephaly and concomitant brachycephaly, and left-sided and right-sided plagiocephaly. The association between severity of plagiocephaly and neurocognitive outcome was assessed through a residualized change approach. RESULTS: There were no significant differences in neurocognitive outcomes between the helmeted and nonhelmeted DP cohorts or the unilateral plagiocephaly and brachycephaly cohorts. Participants with left-sided DP had significantly lower motor coordination scores than participants with right-sided DP (84.8 versus 92.7; effect size = -0.50; P = 0.03). There was a significant laterality by cephalic index interaction, with a negative association between cephalic index and reading comprehension and spelling for participants with left-sided DP. No significant associations were found between severity of presenting or posttreatment deformity and neurocognitive outcome. CONCLUSIONS: Pretreatment and posttreatment severity of plagiocephaly were not correlated with neurocognitive function at school age. Helmet therapy was not associated with better or worse long-term neurocognitive function. However, participants with left-sided DP demonstrated worse neurocognitive outcomes than participants with right-sided DP in the domains of motor coordination and some types of academic achievement. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Three-Dimensional Quantification of Facial Asymmetry in Children with Positional Cranial Deformity.

BACKGROUND: The extent of facial involvement in positional plagiocephaly is only little investigated so far. Investigation methods that take into account the challenging anatomical conditions and growth of infants' faces are desirable. In this study, the authors established a new three-dimensional photogrammetry quantification method evaluating pretherapeutic and posttherapeutic facial asymmetry in positional plagiocephaly. Furthermore, a facial asymmetry index was established and evaluated. METHODS: Three-dimensional photographs of 100 children undergoing treatment with head orthoses were analyzed by constructing a standardized interindividual coordinate system. Defining landmarks, section planes, and point coordinates with a computer-aided design software, both sides of the faces were compared. Facial asymmetry was quantified by measuring differences between left and right sides and pretherapeutic and posttherapeutic changes in each patient. The facial asymmetry index was calculated by putting the absolute differences in relation with the coordinates of the nonaffected side. RESULTS: Present results indicate that positional plagiocephaly results in a distinct facial asymmetry (range, -3.8 to 9.6 mm) in nearly all spatial directions and facial regions. Helmet therapy led to a significant reduction (p < 0.05) of intraindividual facial asymmetry (median change in facial asymmetry index, -1.9 to 3.1 percent). However, no correlation of the Cranial Vault Asymmetry Index and facial asymmetry (Spearman rank correlation coefficient, ρ = -0.09 to 0.47) has been seen. According to these results, severe occipital deformation does not necessarily provoke distinct facial asymmetry. CONCLUSIONS: Present three-dimensional photogrammetry method allows the longitudinal quantification of facial involvement in positional plagiocephaly. Asymmetry has been determined in all facial regions. The facial asymmetry decreased through helmet therapy but was not eliminated completely.

Periocular Asymmetry in Infants with Deformational Posterior Plagiocephaly.

PURPOSE: To analyze the clinical significance of the periorbital features associated with the facial asymmetry that is common in deformational posterior plagiocephaly (DPP). PATIENTS AND METHODS: We identified 32 patients with DPP, photographed their faces and tops of their head, and performed a complete eye examination. Four examiners analyzed the patient's periorbital features on the photographs. RESULTS: Median age was 6.5 months (range 3-12 months). Pseudoptosis was identified in 30 patients and pseudo-brow ptosis in 19. Pseudoptosis was marked in 17 patients. Five patients were misdiagnosed with congenital blepharoptosis and received regular follow-ups for amblyopia checks until the diagnosis of pseudoptosis was established. All patients had normal levator function and symmetric eyelid crease. One patient with pseudoptosis and physiologic anisocoria was diagnosed with pseudo-Horner syndrome after a negative 10% cocaine test. None of the patients developed meridional or occlusion amblyopia. CONCLUSION: DPP is the most frequent form of skull deformation in infants. Its main features are occipital flatness and facial asymmetry. Infants with DPP may present with pseudoptosis and pseudo-brow ptosis on the contralateral side of the occipital flatness. The pseudoptosis in DPP is non-amblyogenic, therefore, ophthalmologic intervention and regular follow-ups are not necessary unless other abnormalities co-exist.

Head orthosis therapy in positional plagiocephaly: longitudinal 3D-investigation of long-term outcomes, compared with untreated infants and with a control group.

Background: As there are very few long-term studies on the effects of head orthosis on deformational plagiocephaly (DP), we investigated the outcomes of patients, including facial symmetry and dental occlusion. Methods: Forty-five infants with DP [cranial vault asymmetry index (CVAI) > 3.5 per cent] were divided into two groups: one treated with head orthosis (32 infants) and another without (13 infants). Another group without head asymmetry (CVAI ≤ 3.5 per cent) served as control. Using 3D-stereophotogrammetry, cranial asymmetry was analysed using symmetry-related variables [CVAI, posterior cranial asymmetry index (PCAI), and ear offset]. Data acquisition was performed before (T1) and at the end of treatment (T2), and at the age of 4 years (T3) for the treated group and at T1 and T3 for the remaining groups. Parameters of facial symmetry and dental occlusion were assessed at T3 for infants with DP. Results: Symmetry-related variables (∆T1-T3) improved significantly more in the treated than the control group, whereas these parameters did not differ significantly between the untreated and control group. Comparing the treated and untreated groups between T1 and T3, the reduction in the asymmetry at the treated group was significantly higher for the CVAI and PCAI. In follow-up, the untreated group showed higher incidences of facial asymmetries than the treated group. Seventy-five per cent of all lateral crossbites found in patients with DP were contralateral to the posterior flattening. Limitations: Due to ethical reasons, the investigation is a non-randomized study. Parameters of facial symmetry were only assed for the treated and untreated groups. Conclusion: Head orthosis therapy in patients with DP leads to significantly better long-term outcomes. Facial asymmetries are more frequent in patients with DP who do not receive this treatment.

Analysis of the correlation between deformational plagiocephaly and neurodevelopmental delay.

BACKGROUND: Deformational plagiocephaly (DP) refers to cranial asymmetry resulting from uneven external forces. A strong association exists between DP and developmental delay. We investigated the effect of DP severity on developmental delay. METHODS: Between 2010 and 2016, data from 155 patients with DP were reviewed retrospectively. Two indices were used to evaluate the deformation quantitatively: cranial index (CI) and cranial vault asymmetry index (CVAI). The Bayley Scales of Infant Development-II was used to evaluate the neurodevelopment of patients. RESULTS: According to the CI of the study population, 2 patients showed scaphocephaly, 12 showed mesocephaly, and 141 showed brachycephaly. For CVAI, 10 patients showed values of <3.5, 10 patients showed mild deformity (3.5-6.25), 27 patients showed moderate deformity (6.25-8.75), and 108 patients showed severe deformity. The means of the mental development index (MDI) and psychomotor development index (PDI) were 91.69 ± 16.8 and 92.28 ± 17.59, respectively; after the exclusion of patients with confounding factors, the values were 96.26 and 92.9, respectively. The Spearman correlation coefficients between MDI and CI and CVAI were -0.019662 and 0.118916, respectively, whereas for PDI, the values were -0.195428 and -0.012386, respectively. CONCLUSIONS: There was a statistically significant neurodevelopmental delay in patients with DP. However, accelerated neurodevelopment was also encountered in many patients. MDI was found to be more affected by multiple confounding factors than PDI, whereas PDI was only affected by congenital anomalies. There was no definitive relationship between the severity of DP and the degree of developmental delay in our study group.

Does shape affect function? Articulatory skills in babbling of infants with deformational plagiocephaly.

PURPOSE: The purpose of this study was to quantitatively analyse pre-speech/early language skills in healthy full-term infants with moderate or severe deformational plagiocephaly (DP) and in infants without any skull asymmetry. METHODS: At 6 and 12 months, 51 children with DP (41 moderate, 10 severe cases) were studied, along with 15 infants serving as control. Deformational plagiocephaly (DP) was objectively determined based on cranial vault asymmetry (CVA) using 3D stereophotogrammetry (3dMDhead System® and Analytics 4.0, Cranioform®). Articulatory skills in babbling were assessed using the articulatory skill (ART-index) and mean syllable number (MSN). At 12 months, standardized parental questionnaires were used to evaluate early language outcomes. RESULTS: Overall, 3546 vocalizations were studied. Statistical tests did not reveal any significant differences of the ART-index between the three groups (ANOVA, F[2,63] = 0.24, p = 0.24). MSN likewise did not differ between the three shape groups (Kruskal-Wallis, p = 0.84). Among the children assigned to the at-risk group for language outcomes at 12 months were seven members of the symmetrical shape group (vs. seven assigned to the normally developing group), nine of the moderate DP group (vs. 27), and one of the severe DP group (vs. six). Fisher's exact test was used to analyse whether helmet therapy in the moderate DP group affected the results by influencing language outcomes, but did not reveal any significant influence (p = 0.712). CONCLUSIONS: The results of this study do not support arguments suggesting that DP is a cognitive risk condition. The suggestion that a direct neurophysiological relationship exists between a DP condition and a cognitive developmental delay remains controversial.

Facial asymmetry correction with moulded helmet therapy in infants with deformational skull base plagiocephaly.

PURPOSE: The recommendation issued by the American Academy of Pediatrics in the early 1990s to position infants on their back during sleep to prevent sudden infant death syndrome (SIDS) has dramatically reduced the number of deaths due to SIDS but has also markedly increased the prevalence of positional skull deformation in infants. Deformation of the base of the skull occurs predominantly in very severe deformational plagiocephaly and is accompanied by facial asymmetry, as well as an altered ear position, called ear shift. Moulded helmet therapy has become an accepted treatment strategy for infants with deformational plagiocephaly. The aim of this study was to determine whether facial asymmetry could be corrected by moulded helmet therapy. MATERIALS AND METHODS: In this retrospective, single-centre study, we analysed facial asymmetry of 71 infants with severe deformational plagiocephaly with or without deformational brachycephaly who were undergoing moulded helmet therapy between 2009 and 2013. Computer-assisted, three-dimensional, soft-tissue photographic scanning was used to record the head shape before and after moulded helmet therapy. The distance between two landmarks in the midline of the face (i.e., root of the nose and nasal septum) and the right and left tragus were measured on computer-generated indirect and objective 3D photogrammetry images. A quotient was calculated between the two right- and left-sided distances to the midline. Quotients were compared before and after moulded helmet therapy. Infants without any therapy served as a control group. RESULTS: The median age of the infants before onset of moulded helmet therapy was 5 months (range 3-16 months). The median duration of moulded helmet therapy was 5 months (range 1-16 months). Comparison of the pre- and post-treatment quotients of the left vs. right distances measured between the tragus and root of the nose (n = 71) and nasal septum (n = 71) revealed a significant reduction of the asymmetry (Tragus-Nasion-Line Quotient: 0.045-0.022; p < 0.0001; Tragus-Subnasale-Line Quotient: 0.045-0.021; p < 0.0001). The control group without treatment showed no significant change in the quotient (Tragus-Nasion-Line Quotient no helmet: 0.049-0.055/Tragus-Subnasale-Line Quotient no helmet: 0.039-0.055). CONCLUSION: Moulded helmet therapy can correct facial symmetry in infants with deformational plagiocephaly and associated facial and basal skull asymmetry.

Nonsynostotic Posterior Brachycephaly with Hindbrain Herniation.

BACKGROUND: Positional plagiocephaly is the most common cause of cranial asymmetry. The underlying cause of Chiari-1 malformation has many possible theories, and anecdotally some pediatric neurosurgeons have had experience of severe cases of positional brachycephaly with Chiari-1. However, to date, there have been no published cases linking nonsynostotic plagiocephaly with Chiari-1 malformation. CASE DESCRIPTION: An 18-month-old boy presented with a head injury. On examination he had a Glasgow Coma Score of 15 with no focal neurologic deficits, but he was noted to have marked posterior brachycephaly. A computed tomography scan showed a slim left-sided hemispheric acute subdural hematoma with no mass effect, which was treated conservatively. Of note, all of his cranial vault sutures were open, and a diagnosis of incidental positional plagiocephaly was made. Subsequent magnetic resonance imaging as part of a work-up to exclude nonaccidental injury showed a small posterior fossa with a steep tentorium and herniation of the cerebellar tonsils to the level of the body of the second cervical vertebra. CONCLUSIONS: Chronic hindbrain herniation is well reported in cases of craniosynostosis, but to our knowledge this is the first published case associated with nonsynostotic deformational plagiocephaly. We hypothesize that severe posterior plagiocephaly can cause disproportion of the posterior fossa: hindbrain volume ratio and acquired chronic cerebellar herniation. Nevertheless, positional plagiocephaly and Chiari-1 are common entities, and it is possible that the dual diagnoses were coincidental in this case. This report serves to raise awareness of a putative causal relationship between positional plagiocephaly, reduced posterior fossa volume, and hindbrain herniation.

Malocclusion in the primary dentition in children with and without deformational plagiocephaly.

OBJECTIVES: Asymmetries of the jaw and orthodontic abnormalities are suspected as long-term consequences of positional cranial deformity. But only few data exist on this issue. As plagiocephaly is a common problem in infancy, potential functional impairments should be investigated to initiate appropriate measures if necessary. The aim of our study was to compare the orthodontic situation in primary dentition of children with positional plagiocephaly and children without cranial deformities. MATERIAL AND METHODS: Fifty children treated by helmet therapy for plagiocephaly and 50 non-affected children (age 1.98-5.69 years) were examined in a cross-sectional study. Orthodontic parameters of all dimensions were assessed and analyzed. RESULTS: Children of the plagiocephalic group showed more often orthodontic alterations compared to the others. Especially the frequencies of a class II malocclusion (36 vs. 14 %), an edge-to edge bite (28 vs. 12 %), and deviations of the midline (38 vs. 16 %) were conspicuous. However, none of the differences was significant (p > 0.003). Of all observed mandibular asymmetries, 69 % appeared as a shift to the contralateral side of the former flattened occipital region. CONCLUSION: Positional head deformity might be associated in some cases with a higher prevalence of occlusal abnormalities in primary dentition. CLINICAL RELEVANCE: Positional plagiocephaly interfaces medicine and dentistry. As it is a common disorder, this etiology has to be considered in the prevention and therapy of malocclusion.

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).

Craneosinostosis sagital. A propósito de un caso / Sagittal craniosynostosis. A case report

La craneosinostosis es el cierre precoz de una o más suturas craneales. Comprende un amplio espectro de malformaciones craneales, desde el cierre aislado de una sola sutura, al cierre de varias suturas, o asociado a otras malformaciones, constituyendo las craneosinostosis sindrómicas. El pediatra debe distinguir las deformidades posicionales o plagiocefalias posturales, más frecuentes, de las craneosinostosis, que requieren tratamiento quirúrgico y cuyo diagnóstico precoz mejora el pronóstico. Se presenta el caso de un lactante con escafocefalia debida a craneosinostosis aislada (AU)

Craniosynostosis is the premature fusion of one or more of the cranial sutures. It includes a wide spectrum of cranial malformations, from closing of an isolated suture to the closing of several sutures, or associated to other malformations, constituting syndromic craniosynostosis. The pediatrician must distinguish the positional deformities or postural plagiocephaly, more frequent, from the craniosynostosis, that needs surgical treatment and whose early diagnosis improves the prognosis. We present the case of an infant with scafocephaly due to isolated craniosynostosis (AU)

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.

Deficient language acquisition in children with single suture craniosynostosis and deformational posterior plagiocephaly.

PURPOSE: This study examined early language acquisition in children with single suture craniosynostosis (SSC) and in children with deformational posterior plagiocephaly. Our purpose was to determine whether infants with SSC have normal language acquisition at the age of 3 years, and whether infants with deformational posterior plagiocephaly demonstrate parallel development when compared with children with SSC. METHODS: The study population includes 61 infants. Twenty of them had synostosis of the sagittal suture, 12 synostosis of other suture and 29 deformational posterior plagiocephaly. Forty-nine of them were operated on a mean age of 10.6 months, and 12 were non-operated children with deformational posterior plagiocephaly. Language skills of participants were prospectively evaluated at the mean age of 3 years 4 months. RESULTS: About one half of the subjects (49%) had normal linguistic development, 30% had slight developmental problems and 21% had severe disorders in speech-language-related skills. These figures showed the prevalence of severe language disorders to be three times higher in our study population when compared with the general population. Children with sagittal synostosis managed better in all language skills compared with other types of SSC. Defective language development was found in deformational posterior plagiocephaly, both operated and non-operated. CONCLUSIONS: We found a noticeable developmental risk for specific language impairment in children with nonsyndromic SSC, and that the deviant language development is observable already in early infancy. Contrary to previous beliefs, the developmental risk for defective language development in deformational posterior plagiocephaly was found in both operated and non-operated subjects.

Development in toddlers with and without deformational plagiocephaly.

OBJECTIVE: To determine whether the heightened risk of developmental delays seen in infancy in patients with deformational plagiocephaly (DP) continues into the toddler years. DESIGN: Longitudinal study comparing the development of children with and without DP, with assessments in infancy (mean age, 7 months) and at age 18 months. SETTING: Infants with DP were recruited from a large craniofacial center, and unaffected infants were recruited from a research registry. PARTICIPANTS: The study included 227 children with DP and 232 children without previously diagnosed DP. MAIN EXPOSURE: Diagnosis of DP by a craniofacial specialist. MAIN OUTCOME MEASURES: Bayley Scales of Infant and Toddler Development, Third Edition, scores. RESULTS: Toddlers with DP scored lower than did unaffected children on all the scales of the Bayley Scales of Infant and Toddler Development, Third Edition. Motor score differences were smaller and cognitive and language score differences were greater than those observed in infancy. CONCLUSIONS: Toddlers with DP continue to exhibit evidence of developmental delays relative to toddlers without DP. These findings do not necessarily imply a causal relationship between DP and development because children with delays may be more likely to develop DP. Nonetheless, it seems that increased developmental surveillance is warranted in this population.

Positional plagiocephaly: what the pediatrician needs to know. A review.

INTRODUCTION: Positional or deformational plagiocephaly is the most common type of cranial asymmetry in infancy and has become more prevalent after the introduction of the "Back to Sleep" campaign in Western countries. However, the supine position cannot be considered as the only etiologic factor and different predisposing variables have been investigated in the last few years. DISCUSSION: The pediatrician should correctly diagnose this condition and exclude the possibility of craniosynostosis in any child with plagiocephaly in order to optimize management and reduce potential morbidity associated with different conditions other than positional ones. In addition, the pediatrician needs to be able to educate parents on methods to proactively decrease the likelihood of the development of occipital flattening, initiate appropriate management, and make referrals when necessary.

Increased incidence of metopic suture abnormalities in children with positional plagiocephaly.

Metopic craniosynostosis has traditionally been reported to be the third most common form of single-suture synostosis. The purposes of this article were to analyze the relationship between metopic craniosynostosis and positional plagiocephaly and to define more clearly the differences between the changes seen with true suture fusion compared with metopic abnormalities secondary to positional changes. This is an institutional review board-approved retrospective review of three-dimensional computed tomographic scans for abnormalities of the metopic suture in all children treated at our institution for positional plagiocephaly between 1997 and 2007. We also independently reviewed the images of all patients treated for metopic synostosis for evidence of positional plagiocephaly during the same period. Among the positional plagiocephaly group, 39% were noted to have some form of metopic suture abnormality. Of the children treated for metopic craniosynostosis, a much smaller percentage also was noted to have evidence of positional plagiocephaly. Between 1997 and 2007, we treated 93 patients (50%) for sagittal synostosis, 41 patients (22%) for unicoronal, 41 patients (22%) for metopic, 4 patients (2%) for lamdoid, and 7 patients (4%) with multiple-suture involvement. This study demonstrates an increasing trend toward metopic suture abnormalities during the past 10 years, which corresponds to the same time interval of an increased incidence of positional abnormalities. It is postulated that some of these abnormalities are related to deformational forces resulting from posterior pressure. Altered mechanical forces from supine infant positioning may be associated with early metopic suture ridging and dysmorphology distinct from true craniosynostosis.

Accuracy of three-dimensional photogrammetric images in non-synostotic cranial deformities.

OBJECTIVE: Given the increasing incidence of deformational plagiocephaly due to infants' supine sleeping position to prevent sudden infant death syndrome, reliable anthropometric diagnostics are needed. Besides the traditional method of measuring landmarks with callipers, three-dimensional (3D) photography has great potential. In this investigation the accuracy of 3D photogrammetry is studied. METHODS: The study included 100 randomly chosen children between the ages of 4 and 20 months with a non-synostotic cranial deformity in a retrospective analysis. Measurements of diagonals A and B on the infant's head were obtained once using callipers. 3D photographs of these children were measured 5 times by 5 clinicians separately. RESULTS: The inter- and intra-rater agreements of the 3D measurements had low variability in the variance component analysis. The standard deviations for reproducibility and repeatability were 0.117-0.283 cm for diagonals A and B. The intra-class correlation coefficients for the inter-rater reliability resulted in excellent agreement (0.97 for plagiocephaly, 0.98 for brachycephaly, 0.96 for combined deformity). The comparison of the 3D photographic and callipers measurements showed that 3D photography resulted in a slight over-estimation. CONCLUSION: 3D photogrammetry is potentially a reliable tool for treatment planning and follow-up of abnormal head shapes in infancy.

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.