Posterior Plagiocephaly Research Articles

Understanding the heterogenicity of unicoronal synostosis – A morphometric analysis of cases compared to controls

BackgroundPreoperative severity of unicoronal synostosis varies greatly and involves the frontal bone, skull base and orbits. Degree of deformity affects long-term morphological and functional outcome after surgery. The aim of this study was to describe the morphological heterogenicity and investigate its relation to patient-specific factors. Materials and methodsIn this retrospective cohort study, non-syndromic unicoronal synostosis patients treated between 2006-2022 at Necker Hospital, France, or Uppsala University Hospital, Sweden, were included and matched to controls. Severity of skull base, orbital and posterior skull asymmetry, degree of anterior plagiocephaly and Harlequin deformity, lateralization, head circumference, age, timing of metopic fusion and fusion of peri-pterionic sutures were investigated. ResultsNinety-five patients and ninety-three controls were included. Skull base asymmetry was linearly related to orbital asymmetry (p < 0.001), correlated with earlier CT scans (p = 0.004), anterior (p < 0.001) and posterior (p = 0.03) plagiocephaly. Posterior plagiocephaly was more common in patients (31%) compared with controls (5%) (p < 0.001). A patent metopic suture above nine months of age was associated with severe Harlequin deformity (p = 0.04) and lower head circumference when fused (p = 0.03). Fronto-sphenoidal suture fusion was associated with later CT scans (p < 0.001) and less skull base asymmetry (p = 0.002). Spheno-parietal fusion was correlated with decreased skull base asymmetry. Right lateralization was more common in females. ConclusionHeterogenicity of unicoronal synostosis seems to be predominantly explained by variability in skull base morphology. Peri-pterionic fusions might limit deformity.

Prevalence and Severity of Positional Posterior Plagiocephaly and Positional Posterior Brachycephaly in Children and Adolescents in Japan.

This study aimed to assess the prevalence, severity, and natural history of positional posterior plagiocephaly (PPP) and positional posterior brachycephaly in Japan. We conducted a cross-sectional study of pediatric patients, ranging from 0 to 15 years old, evaluated for head trauma with negative computed tomography (CT) findings. The cranial vault asymmetry index (CVAI) was calculated using CT images at the superior orbital rim. Asymmetry according to CVAI values was subcategorized as follows: mild (3.5%-7%), moderate (7%-12%), and severe (>12%). The results were analyzed according to different age groups: group 1, 2-23 months (54 patients); group 2, 2-6 years (123 patients); and group 3, 7-15 years (123 patients). Overall, 300 patients were included (109 [36.3%] girls and 191 [63.7%] boys). The overall prevalence of PPP in the 300 patients was 46.7% (140 patients). PPP prevalence decreased consistently with age group: group 1, 57.4%; group 2, 47.2%; and group 3, 41.5%. Severe asymmetry was seen in all age groups. The overall mean cephalic index (CI) was 85.2. Cephalic index scores decreased consistently with age: group 1, 87.4; group 2, 85.1; and group 3, 84.3. The prevalence of PPP in Japan was higher than that reported in other countries. Although there was an overall decrease in the prevalence and severity of PPP with increasing patient age, PPP does not necessarily resolve spontaneously in all children. Furthermore, severe asymmetry was seen across all age groups.

Computed tomography quantitative analysis of cranial vault dysmorphology and severity of facial complex changes in posterior synostotic plagiocephaly patients.

Posterior synostotic plagiocephaly (PSP) impacts craniofacial skeleton. Study quantifies facial changes in children with PSP to investigate the impact of age and PSP severity at diagnosis on the facial dysmorphology. High-resolution preoperative CT images of 22 infants with PSP were analyzed. They were divided according to the early or late age at time of diagnosis. Each group was further subdivided according to the severity of PSP evaluated by the cranial vault asymmetry index (CVAI): mild-moderate PSP (CVAI between 3 and 12%) and severe PSP (CVAI > 12%). Analysis of the facial complex was performed. Each group was compared with age-matched healthy subjects. All children exhibited unilateral lambdoid suture synostosis. The "early" diagnosis group consisted of 7 children with mild-moderate PSP while the "late" diagnosis group of 15 children in which 6 children had mild-moderate and 9 children severe PSP. All children showed altered position of glenoid fossae and mandibular asymmetry characterized by reduced mandibular diagonal distance length on the affected side while the subgroup of children with severe PSP detected in "late" diagnosis group had also altered mandibular inclination and reduced midfacial depth on both sides. PSP causes cranial base dysmorphology which drives changes in facial complex growth; the severity of facial changes mainly depends on the severity of cranial vault dysmorphology detected by CVAI. Mandible reshapes early under the stress of altered biomechanical forces of the skull base while changes in the maxilla are secondary to the asymmetric growth of the mandible and occur only in severe cases.

Evaluation of the Effects of Helmet Therapy on Head Deformities: A Systematic Review of Literature

Plagiocephaly is a common deformity of the skull. Various treatment approaches have been used for the subjects with head deformities, including use of helmet orthosis, repositioning, exercise, and only observation. The aim of this review was to check the efficiency of helmet therapy compared to other available approaches and also to check the effects of some parameters such as the duration of use, and age of the subjects on treatment of the subjects with this deformity. A search was done in some databases, including Google scholar, ISI Web of knowledge, PubMed, and Scopus. Some keywords such as plagiocephaly, posterior plagiocephaly, and occipital plagiocephaly were used in combination with positional modeling, helmet therapy, helmet orthosis, exercise, and observation. The quality of the studies was evaluated by use of Down and Black tool. Twenty-four studies were found on the mentioned keywords. The quality of the studies varied between 11 and 21. Although the efficiency of helmet therapy was more than other treatment approaches, the final outputs depend on optimal time and deformity criteria. This approach is effective, well tolerated with no morbidity. Moreover, it does not influence on the quality of life of the subjects. The efficiency of helmet therapy to correct head deformities is more than other available approaches. However, its effects depend on the age of the subjects at onset of treatment, duration of treatment, and severity of the deformity. Any side effects of this treatment method can be decreased by proper education of the subjects.

Facial asymmetry in children with either unilateral lambdoid craniosynostosis or positional posterior plagiocephaly.

In unilateral lambdoid craniosynostosis (ULC), the posteriorly situated lambdoid suture of the cranial vault fuses prematurely. Positional posterior plagiocephaly (PPP) causes flattening of the posterior side of the head, either through external forces or through underlying differences in brain development. Both conditions cause occipital flattening of the head, but the aetiology is different. Eight ULC children were compared with 16 sex- and age-matched PPP children. 3D computer tomography scans of all 24 children were analysed with Dolphin imaging software. The location and symmetry of the temporomandibular joint (Co), and the symmetry of the maxillary anterior nasal spine (ANS) and the mandibular symphysis (Pgn) were analysed. Furthermore, the mandibular bone (Co-Pgn) length, corpus length, ramus height, positional changes in the external acoustic meatus (PoL) and the distance from the orbital margin to the articular fossa were measured. In all eight ULC children, the Co was anteriorly displaced on the affected side compared with the unaffected side. In all ULC and PPP children, the ANS, which is considered the bony maxillary midpoint, was shifted towards the affected side. In all ULC children, the mandibular bone (Co-Pgn) was shorter on the affected side. The PoL was antero-inferiorly positioned in all ULC children on the affected side compared with the unaffected side. Our results show that both types of posterior plagiocephaly are associated with an asymmetric position of the Co and asymmetry of the mandible and maxilla. Facial asymmetry was more frequently seen in ULC than PPP children.

Rotation Advancement Distraction Osteogenesis Technique is Attributed to Unilateral Lambdoid Synostosis and is Used to Correct Posterior Plagiocephaly.

Unilateral lambdoid synostosis is the rarest form of craniosynostosis. Surgical correction is technically challenging from a morphological standpoint and is used to decompress intracranial pressure. Herein, the authors present a "rotational advancement bone flap" technique for the repair of posterior plagiocephaly. A bicoronal skin incision was performed, to expose the occipital region. The rotational advancement bone flap was designed to hinge off the nonsynostotic side, and it was set up to include an occipital protrusion on the normal side. On the premature side, an advancement bone flap was set up to include the flattened and deformed occipital bone. A titanium cranial distractor, which was attached between the bone flap and the anterior bone. An absorbable plate, which would later become a hinge due to the movement of the bone flap, was attached to the area for reinforcement. The authors technique was able to decompress intracranial pressure with satisfactory esthetic outcomes.

UCSQ Method Applied on 3D Photogrammetry: Non-Invasive Objective Differentiation Between Synostotic and Positional Plagiocephaly.

Objective differentiation between unilateral coronal synostosis (UCS) and positional posterior plagiocephaly (PPP) based on 3D photogrammetry according to Utrecht Cranial Shape Quantificator (UCSQ). Retrospective study. Primary craniofacial center. Thirty-two unoperated patients (17 UCS; 15 PPP) (age < 1 year). Extraction of variables from sinusoid curves derived using UCSQ: asymmetry ratio forehead and occiput peak, ratio of gradient forehead and occiput peak, location forehead and occiput peak. Variables, derived using 3D photogrammetry, were analyzed for differentiation between UCS and PPP. Frontal peak was shifted to the right side of the head in left-sided UCS (mean x-value 207 [192-220]), and right-sided PPP (mean x-value 210 [200-216]), and to the left in right-sided UCS (mean x-value 161 [156-166]), and left-sided PPP (mean x-value 150 [144-154]). Occipital peak was significantly shifted to the right side of the head in left-sided PPP (mean x-value 338 [336-340]) and to the left in right-sided PPP (mean x-value 23 [14-32]). Mean x-value of occipital peak was 9 (354-30) in left- and 2 (350-12) in right-sided UCS. Calculated ratio of gradient of the frontal peak is, in combination with the calculated asymmetry ratio of the frontal peak, a distinctive finding. UCSQ objectively captures shape of synostotic and positional plagiocephaly using 3D photogrammetry, we therefore developed a suitable method to objectively differentiate UCS from PPP using radiation-free methods.

Wormian bones and nonsyndromic craniosynostosis: A comparative study using computed tomography

Introduction: Wormian bones (WB) are accessory bones positioned within the sutures or fontanelles. Although several studies have associated them with genetic disorders, ethnicity, and skull deformations, their relationship with nonsyndromic craniosynostosis (CS) has not been established, especially among Brazilians. Therefore, this study was conducted to analyze the incidence and association of WB with nonsyndromic CS in Brazilian children.&#x0D; Methods: An observational and retrospective study was conducted using computed tomography with 3D reconstruction in Brazilian children aged &lt;3 years to compare the incidence and number of WB between regular children (group 1) and those with nonsyndromic CS (group 2).&#x0D; Results: A total of 140 children, comprising 62.9% boys (p &lt; 0.001) with a mean age of 8.78 months, were included in this study. The most common types of CS were trigonocephaly (34; 48.6%), scaphocephaly (25; 35.7%), anterior plagiocephaly (5; 7.1%), posterior plagiocephaly (3; 4.2%), and brachycephaly (3; 4.2%). WB were more common in regular children (41; 58.6%) than in children with CS (28; 40.0%) (p = 0.028). Regular children had an average of 2.12 WB versus 1.32 WB in children with nonsyndromic CS (p = 0.024).&#x0D; Conclusion: The significantly less incidence of WB in Brazilian children with nonsyndromic CS was quite different from that reported in other studies and could indicate the tendency of this disease to have disturbance in ossification of the cranial vault, the impact of ethnicity, and probably the lack of additional compensatory skull growth.&#x0D; &#x0D;

Application of Deep Learning Techniques for Automated Diagnosis of Non-Syndromic Craniosynostosis Using Skull.

Non-syndromic craniosynostosis (NSCS) is a disease, in which a single cranial bone suture is prematurely fused. The early intervention of the disease is associated with a favorable outcome at a later age, so appropriate screening of NSCS is essential for its clinical management. The present study aims to develop a classification and detection system of NSCS using skull X-ray images and a convolutional neural network (CNN) deep learning framework. A total of 56 NSCS cases (scaphocephaly [ n = 17], trigonocephaly [n = 28], anterior plagiocephaly [n = 8], and posterior plagiocephaly [n = 3]) and 25 healthy control infants were included in the study. All the cases underwent skull X-rays and computed tomography scan for diagnosis in our institution. The lateral views obtained from the patients were retrospectively examined using a CNN framework. Our CNN model classified the 4 NSCS types and control with high accuracy (100%). All the cases were correctly classified. The proposed CNN model may offer a safe and high-sensitivity screening of NSCS and facilitate early diagnosis of the disease and better neurocognitive outcome for patients.

Effectiveness of osteopathic care in the treatment of infant positional posterior plagiocephaly

Introduction.Posterior Positional plagiocephaly (PPP) is a cranial deformation seen in infants. The case number increased since 1992, following the supine sleeping advice. Most of the time several factors are combining together, leading to a multi-level lack of mobility of the infant, thus leading to the cranial deformation. Studies show this deformation to be linked with perturbations in various fields.Objective.This study aims to assess the efficacy of osteopathic care on the severity of the PPP.Materials and methods.100 infants were recruited in an osteopathic practice in Lyon (France). All the infants followed the same study protocol; however, the treatment was individualized. Three consultations occurred with a 15 days′ interval over 30 days′ period. Craniometric measurements were realized with Mimos® craniometer on each consultation.Results.The mean severity of the PPP was 10,1 mm at Day 0. At day 30, this average was down to 6 mm. It is a significant (p&lt;0,001) clinical and statistical decrease. Out of the 100 infants, 98 had a decreased deformation while two stagnated.Conclusions.As every infant is unique, every plagiocephaly is different, thus the treatment was not standardised. This study suggest some effectiveness for osteopathic care in the cranial asymmetry of infants with PPP. Other studies should be done, some with a control group, in order to compare our results with spontaneous evolution of plagiocaphaly.

A Neonate with an Abnormally Shaped Head

A female newborn has an abnormally shaped head (Figs 1 and 2). Figure 1. Profile of infant. Figure 2. View of top of infant’s head. ### Prenatal and Birth Histories ### Presentation (Day 1) The infant’s respiratory distress resolved 2 hours after birth and she transitioned off CPAP. Dysmorphic features were noted on physical examination at 2 days of age and hence, further evaluation was undertaken. ### Vital Signs ### Physical Examination (Day 2)

Clinical and Radiological Features of Some Rare Forms of Craniosynostosis in Clinical Practice

There are rare forms of craniosynostosis with single or multiple suture involvement. These are sphenofrontal plagiocephaly; posterior plagiocephaly with unilateral lambdoid synostosis; combined sagittal and metopic synostosis; combined sagittal and unilateral coronal synostosis; and bilateral lambdoid and sagittal synostosis with Mercedes–Benz pattern. In our study, we aimed to present both clinical and three-dimensional computed tomographic characteristics of these rare forms of craniosynostosis.

Ipsilateral coronal and lambdoid synostosis: a case of anterior and posterior plagiocephaly in a child with hypophosphatasia

Ipsilateral coronal and lambdoid synostosis: a case of anterior and posterior plagiocephaly in a child with hypophosphatasia

Distinguishing Craniomorphometric Characteristics of Unilateral Lambdoid Craniosynostosis.

Unilateral lambdoid synostosis (ULS) represents the rarest form of single suture nonsyndromic craniosynostosis. Differentiating between posterior deformational plagiocephaly (DP) and ULS has been difficult due to overlapping clinical findings. Past analyses have been limited by sample size. This study was undertaken to clarify anatomical features of ULS. A multiinstitution study was undertaken, analyzing CT imaging from patients with documented ULS to determine clinical and pathological characteristics. Similar analyses were performed on DP patients to differentiate the 2 conditions. Twenty-seven ULS patient scans and 10 DP scans were included. For ULS patients mean age was 6.6 months, majority male (75%), and majority left-sided ULS (71%). The synostosed side ear was anteriorly displaced in 100% of ULS patients, mean difference of 9.6° (P < 0.001), and inferiorly in 96.3% of patients, mean difference of 4.4 mm, relative to the nonsynostosed side. The posterior fossa deflection (PFD) was deviated 5.9° toward the synostosed side relative to the anterior midline. In DP, there was no significant difference between sides in EAC measurements. The PFD and EAC displacements were significantly smaller in DP relative to ULS (P < 0.001 for each). An ipsilateral mastoid bulge was found in 100% of ULS and 0% of DP on CT imaging. Contrary to some previously published findings, the ear is more anteriorly displaced ipsilateral to the fused lambdoid suture in 100% of ULS patients. Ear position alone is not a reliable indicator to differentiate between DP and ULS. A mastoid bulge is a more reliable indicator of ULS.

Pediatric cranial deformations: demographic associations.

No study has established a relationship between cranial deformations and demographic factors. While the connection between the Back to Sleep campaign and cranial deformation has been outlined, considerations toward cultural or anthropological differences should also be investigated. The authors conducted a retrospective review of 1499 patients (age range 2 months to less than 19 years) who presented for possible trauma in 2018 and had a negative CT scan. The cranial vault asymmetry index (CVAI) and cranial index (CI) were used to evaluate potential cranial deformations. The cohort was evaluated for differences between sex, race, and ethnicity among 1) all patients and 2) patients within the clinical treatment window (2-24 months of age). Patients categorized as "other" and those for whom data were missing were excluded from analysis. In the CVAI cohort with available data (n = 1499, although data were missing for each variable), 800 (56.7%) of 1411 patients were male, 1024 (79%) of 1304 patients were Caucasian, 253 (19.4%) of 1304 patients were African American, and 127 (10.3%) of 1236 patients were of Hispanic/Latin American descent. The mean CVAI values were significantly different between sex (p < 0.001) and race (p < 0.001). However, only race was associated with differences in positional posterior plagiocephaly (PPP) diagnosis (p < 0.001). There was no significant difference in CVAI measurements for ethnicity (p = 0.968). Of the 520 patients in the treatment window cohort, 307 (59%) were male. Of the 421 patients with data for race, 334 were Caucasian and 80 were African American; 47 of the 483 patients with ethnicity data were of Hispanic/Latin American descent. There were no differences between mean CVAI values for sex (p = 0.404) or ethnicity (p = 0.600). There were significant differences between the mean CVAI values for Caucasian and African American patients (p < 0.001) and rate of PPP diagnosis (p = 0.02). In the CI cohort with available data (n = 1429, although data were missing for each variable), 849 (56.8%) of 1494 patients were male, 1007 (67.4%) of 1283 were Caucasian, 248 (16.6%) of 1283 were African American, and 138 patients with ethnicity data (n = 1320) of Hispanic/Latin American descent. Within the clinical treatment window cohort with available data, 373 (59.2%) of 630 patients were male, 403 were Caucasian (81.9%), 84 were African American (17.1%), and 55 (10.5%) of 528 patients were of Hispanic/Latin American descent. The mean CI values were not significantly different between sexes (p = 0.450) in either cohort. However, there were significant differences between CI measurements for Caucasian and African American patients (p < 0.001) as well as patients of Hispanic/Latin American descent (p < 0.001) in both cohorts. The authors found no significant associations between cranial deformations and sex. However, significant differences exist between Caucasian and African American patients as well as patients with Hispanic/Latin American heritage. These findings suggest cultural or anthropological influences on defining skull deformations. Further investigation into the factors contributing to these differences should be undertaken.

Diagnosis, Management, and Outcome in 9 Children with Unilateral Posterior Synostotic Plagiocephaly

Posterior synostotic plagiocephaly represents a rare challenging type of craniosynostosis, often misdiagnosed as a simple posterior positional plagiocephaly. Underdiagnosed forms may result in delayed diagnosis and neurosurgical management, with potential ophthalmologic, cognitive, and aesthetic sequelae in children. Here we retrospectively analyzed data of 9 posterior synostotic plagiocephaly infants treated in our center over a 10-year period (January 2000-December 2009). Patients averaged 10 months of age, and there was a clear male predominance (8 males/1 female). Cerebral computed tomography scan was performed in all patients, and the abnormal suture was located at the right side in 7 cases (77.77%). Ipsilateral occipitomastoid bulge and occipital flattening were found in all cases (100%). Posterior ear displacement was found in 7 cases (77.77%), and there was minimal facial asymmetry in 4 cases (44.44%). The ophthalmoscopic examination found a papillary edema grade 1 in 2 cases and grade 2 in 4 cases. There were no deaths or reoperation in our series. All of our patients underwent a neurosurgical correction, with total ophthalmic recovery in all patients with preoperative papillae edema. Eight patients had an early satisfactory aesthetic aspect. Long-term follow-up in treated children found a normal childhood, with no major school dysfunctions and normal social integration. Interesting findings in our series were male gender predominance and predilection of the right-side synostotic lambdoid suture. We think that early correct diagnosis and appropriate neurosurgical treatment may prevent potential complications such as neurocognitive and aesthetic sequelae in children with posterior synostotic plagiocephaly.

Positional posterior plagiocephaly: a single-center review.

The authors sought to assess the prevalence and severity of positional posterior plagiocephaly (PPP) in the pediatric population at a tertiary care center. The authors conducted a retrospective review of 1429 consecutive patients aged 2 months to 18 years who presented with head trauma and a negative CT scan in 2018. The cohort was stratified by age. The cranial vault asymmetry index (CVAI) was calculated at the superior orbital rim. Asymmetry was categorized according to the following CVAI scores: mild (3.5%-7%), moderate (7%-12%), and severe (> 12%). Patients were grouped by age to assess PPP at different stages of head development: group 1, 2-5 months; group 2, 6-11 months; group 3, 12-23 months; group 4: 2-4 years; group 5, 5-8 years; group 6, 9-12 years; and group 7, 13-18 years. Patients with a history of shunted hydrocephalus, craniosynostosis, skull surgery, or radiographic evidence of intracranial trauma were excluded. The overall cohort prevalence of PPP was 24.8% (354 patients). PPP prevalence was higher among younger patients from groups 1-3 (40.4%, 33.5%, and 0.8%, respectively). There was a continued decline in PPP by age in groups 4-7 (26.4%, 20%, 20%, and 10.8%, respectively). Mild cranial vault asymmetry was noted most often (78.0%, 276 patients), followed by moderate (19.5%, 69 patients) and severe (2.5%, 9 patients). There were no patients in group 6 or 7 with severe PPP. PPP is prevalent in pediatric populations and most commonly presents as a case of mild asymmetry. Although there was an overall decline of PPP prevalence with increasing age, moderate asymmetry was seen in all age groups. No patients in the cohort had severe asymmetry that persisted into adolescence.

Isolated Lambdoid Craniosynostosis.

Lambdoid craniosynostosis is an extremely rare anomaly in which there is premature fusion of one or both lambdoid sutures. The mainstay of treatment is surgical intervention, for which various procedures have been described, but there is a paucity of data on long-term outcomes. This study examines the long-term outcomes in the surgical management of this challenging condition, showing that accurate diagnosis and careful planning can lead to safe and consistent results. A retrospective chart review was performed looking at all cases of isolated lambdoid craniosynostosis treated with surgical intervention by the senior author from 1999 to 2016. Data collected included gender, age at diagnosis, age at surgery, length of follow up, method of diagnosis, side of affected suture, pre-operative and post-operative physical exam findings, surgical technique, complications, re-operation rate, and associated torticollis. Twenty-five patients (N = 25) were included in the study. All patients underwent posterior calvarial remodeling with/without barrel stave osteotomies and full thickness calvarial bone grafts. Mean length of follow up after operative intervention was 43.8 months (+/- 23.2 months). All patients were judged to have significantly improved head contour which was near-normal at conversational distance during post-operative follow up by the senior author. Residual plagiocephaly was present in 24% of patients. There were no major complications in this series. Reoperation rate was 8%. Seventy-six percent of patients also presented with torticollis, of which 37% had refractory torticollis that required sternocleidomastoid (SCM) release by the senior author. The authors present one of the largest series of operative cases of isolated lambdoid craniosynostosis to date. Our data show that with accurate diagnosis and careful planning, safe and consistent long-term results can be achieved with surgical intervention. A significant number of patients in our series also presented with concomitant torticollis. The authors recommend that all patients being evaluated for posterior plagiocephaly should also be evaluated for torticollis, because without recognition and intervention, patients may continue to have residual facial asymmetry and head shape abnormalities despite optimal surgical correction of the lambdoid synostosis.

Reduction Cranioplasty Using a Three-dimensional Preoperative Virtual Model for Treatment of Extreme Macrocephaly and Positional Plagiocephaly Secondary to Untreated Hydrocephalus - Case Description and Literature Review

Abstract INTRODUCTION Severe macrocephaly may occur due to untreated hydrocephalus and may persist even after treatment. Reductive cranioplasty intends to reduce the infant's head size to optimize motor development. Few surgical techniques of reductive cranioplasty have been reported. We present an innovative technique in which a 3D printed skull model was used to plan the surgery. METHODS A 21-mo-old male from Cape Verde was admitted to our center presenting untreated congenital hydrocephalus, severe macrocephaly and left posterior plagiocephaly. The patient had been initially treated with a right parietal ventriculo-peritoneal shunt placement that was not working for a long time. On admission, his head circumference was 64 cm and he was unable to lift the head up. The shunt was promptly revised but the severe macrocephaly persisted. A reductive cranioplasty was proposed for both functional and cosmetic reasons. The skull was reconstructed from a volumetric CT scan and a 3D volume was printed using a fused deposition modeling printer. This model was then used to simulate the cranioplasty using a total cranial vault remodeling technique. All the removed parts and augmenting cuts of the printed cranial vault were measured and transferred to the patients' skull to replicate the plan. RESULTS The surgical result was very similar to the planned one. One year later, the patient's head circumference was reduced to 59 cm and the cranial deformity had markedly improved. He had acquired head control and was able to sit without support. CONCLUSION Reductive cranioplasty is an off-standard and complex surgery. The authors consider that a preoperative simulation using a 3D computer assisted model may optimize the surgical results.

Prevalence and severity of positional plagiocephaly in children and adolescents.

Though positional posterior plagiocephaly (PPP) is considered common in infants since the pediatric recommendations of "Back to Sleep", several aspects of its natural history still remain unclear. The aim of this study is to understand the actual prevalence and severity of PPP in children and adolescents. Head CT scans performed for head trauma during the period September 2016-September 2017 were retrospectively analyzed in a total of 165 children ranging from 0 to 18years of age (101 boys). Cranial vault asymmetry index (CVAI) was calculated at the level of the superior orbital rim. CVAI values greater 3.5% was considered index of asymmetry. The results were analyzed according to different age groups: group I: 1month to 1year of age (37 children), group II: 2 to 4years (32 children), group III: 5 to 8years (36 children), group IV: 9 to 12years (27 children), and group V: 13 to 18years (33 children) and the severity of asymmetry according to CVAI values: mild group (CVAI range 3.5-7%), moderate group (CVAI range 7-12%), and severe group (CVAI > 12%). The total prevalence of PPP in the 165 children was 25%. While the prevalence in infants of group I was estimated to be 40.5%, it was 15.6% in group II, 30.5% in group III, 18.5% in group IV, and 12% in group V. The mean and maximum degrees of deformation were 3.5% and 15.1%, respectively. Most children had a mild asymmetry. One child (group II) presented a severe asymmetry. The degree of the asymmetry varied according to the groups but moderate asymmetry could be found at all ages even in groups IV and V. This study analyzing PPP in an unselected unbiased pediatric population shows that PPP has a high prevalence in adolescents. It confirms that the prevalence of deformational plagiocephaly is more common than usually reported and that PPP may persist at a late age.