The use of a single-piece bone flap for cranial reshaping in anterior craniosynostosis patients: clinical experience and a description of a novel technique

Background Craniosynostosis is known as premature closure of one or more of the cranial sutures. Anterior craniosynostosis involves anterior plagiocephaly and trigonocephaly. One of the issues in anterior craniosynostosis skull reshaping is maintaining an aesthetically pleasing forehead curve. Therefore, in this article, we demonstrate our novel technique to use a single-piece bone flap for cranial reshaping of the anterior mold in patients diagnosed with anterior craniosynostosis. A retrospective record review of patients who underwent single piece bone flap cranial reshaping for correction of unicoronal synostosis (UCS) and metopic synostosis (MS) at an Academic Institute in Riyadh, Saudi Arabia, between 2018 and 2020, was conducted. Results Six non-syndromic consecutive patients were included. Three of the patients had MS. The mean age at surgery was 11.16 months (range, 6–19 months). The average OR time was 315 min (range, 263–368 min). The average intraoperative blood loss was 225 ml (range, 100–400 ml). All patients had achieved acceptable functional and aesthetic results. Conclusion Our novel technique is an innovative and efficient reconstructive technique to simultaneously address MS and UCS and minimize intraoperative bleeding and surgery time. However, more studies with more cases are required.

Craniofacial characteristics and cosmetic satisfaction of patients with sagittal and metopic synostosis: a case–control study using 3D photogrammetric imaging

Purpose The aim of this study was to investigate the craniofacial and aesthetic characteristics of adult metopic and sagittal craniosynostosis patients operated on in early childhood compared to controls. The goal was to find objective measurements that would correlate with the patient’s subjective self-evaluation of their own cosmetic appearance. Methods The study population consisted of 49 patients from whom 41 had premature fusion of the sagittal and in 8 of metopic suture. There were 65 age and gender matching controls from The Finish National Register. The 3D photogrammetric models were created from all patients and controls. The images were analysed using Rapidform 2006. Facial landmarks were set by the standard Farkas points. Facial symmetry parameters were calculated by using the landmarks and the mirror shell of the face. Aesthetic evaluation was done from standard photographs using panels. Subjective satisfaction with one’s own appearance was evaluated using questionnaires. Results Patients had the greatest asymmetry in the forehead area when compared to controls (symmetry percentage 59% versus 66%, p = 0.013). In the control group, the gap between the eyes was smaller than in the case group, resulting in an absolute 2 mm difference (p = 0.003). The area of the chin and the landmarks were more located on the left side in the patient group, resulting in up to a 1.1 mm difference between the groups (p = 0.003). Only a weak association was found between craniofacial symmetry and appearance evaluations. Conclusion Patients operated on because of sagittal and metopic synostoses were found to have facial asymmetry at long follow-up. However, the differences were < 3 mm and not clinically important. The long-term aesthetical outcome of the surgery performed because of sagittal or metopic craniosynostosis based on the 3D image evaluation was good.

A Three-Dimensional-Based Morphometric Analysis of a Standardized Overcorrection Technique for Fronto-Orbital Advancement in Metopic Craniosynostosis

Introduction The concept of “overcorrection” for trigonocephaly has been reported to achieve both anterior cranial fossa expansion and normalization of craniofacial form. The purpose of this study is to describe in detail a standardized technique to fronto-orbital advancement utilizing the concept of “overcorrection” and objectively evaluate intermediate results. Methods This retrospective study included patients with isolated metopic synostosis who underwent surgery via the proposed surgical technique and age and sex-matched unaffected controls. Craniofacial morphometric analysis was performed on pre-, immediate post-, and intermediate postoperative (>2 years) three-dimensional (3D)-rendered computed tomographic (CT) scans and photographs. Key CT-based measurements included interzygomaticofrontal suture distance (IZFS), endocranial bifrontal angle (ECA), and temporal expansion. 3D photogrammetry was performed using established measurements and associated Z-scores converted. A Paired t-test and analysis of variance were performed when appropriate. Results Forty-one patients were included. A comparison of pre- and immediate postoperative CT scans demonstrated statistically significant increases in all measurements. Subset analysis of 12 patients with intermediate follow-up (age: 39.6 ± 3.6 months) demonstrated significant differences from preoperative values except for IZFS, which decreased from immediate postoperative values and was smaller than age- and sex-matched controls. 3D photogrammetry demonstrated a mean Z-score above the norm for frontal breath. 3D photogrammetry is also positively correlated with CT-based measurements. Conclusions This standardized “overcorrection” approach for trigonocephaly can provide the appropriate changes to maintain a normal ECA despite a reduction in bifrontal width over time. 3D photogrammetry positively correlated with CT-based measurements and may provide useful information when following patients clinically. Long-term follow-up assessment to determine the necessary degree of overcorrection at skeletal mature is needed.

“Validation of Artificial Intelligence Severity Assessment in Metopic Craniosynostosis”

Objective Several severity metrics have been developed for metopic craniosynostosis, including a recent machine learning-derived algorithm. This study assessed the diagnostic concordance between machine learning and previously published severity indices. Design Preoperative computed tomography (CT) scans of patients who underwent surgical correction of metopic craniosynostosis were quantitatively analyzed for severity. Each scan was manually measured to derive manual severity scores and also received a scaled metopic severity score (MSS) assigned by the machine learning algorithm. Regression analysis was used to correlate manually captured measurements to MSS. ROC analysis was performed for each severity metric and were compared to how accurately they distinguished cases of metopic synostosis from controls. Results In total, 194 CT scans were analyzed, 167 with metopic synostosis and 27 controls. The mean scaled MSS for the patients with metopic was 6.18 ± 2.53 compared to 0.60 ± 1.25 for controls. Multivariable regression analyses yielded an R-square of 0.66, with significant manual measurements of endocranial bifrontal angle (EBA) (P = 0.023), posterior angle of the anterior cranial fossa (p < 0.001), temporal depression angle (P = 0.042), age (P < 0.001), biparietal distance (P < 0.001), interdacryon distance (P = 0.033), and orbital width (P < 0.001). ROC analysis demonstrated a high diagnostic value of the MSS (AUC = 0.96, P < 0.001), which was comparable to other validated indices including the adjusted EBA (AUC = 0.98), EBA (AUC = 0.97), and biparietal/bitemporal ratio (AUC = 0.95). Conclusions The machine learning algorithm offers an objective assessment of morphologic severity that provides a reliable composite impression of severity. The generated score is comparable to other severity indices in ability to distinguish cases of metopic synostosis from controls.

Morphologic Severity of Craniosynostosis: Implications for Speech and Neurodevelopment

Background: Single-suture craniosynostosis (SSC) can be associated with neurodevelopmental deficits. We examined the correlation between morphologic severity and incidence of speech-language or psychological concerns. Methods: In 62 patients (33 sagittal, 17 metopic, and 12 unicoronal), morphologic severity was determined via preoperative computed tomography (CT). Severity metrics for sagittal, metopic, and unicoronal synostosis were adjusted cephalic index (aCI), interfrontal angle (IFA), and anterior cranial fossa area ratio (ACFR), respectively. Speech-language and psychological concerns were assessed at age ≥4.5 years and defined as recommendation for therapy or monitoring. Results: Mean assessment age was 5.7 years; 32% had a speech-language concern and 44% had a psychological concern; 44% had neither. Sagittal: Mean aCI of those with a speech-language concern (0.62) and those without (0.62) were equivalent ( P = .580), as were mean aCI of those with a psychological concern (0.62) and those without (0.62; P = .572). Metopic: Mean IFA with (117.9) and without (125.2) a speech-language concern were equivalent ( P = .326), as were mean IFA with (120.2) and without (123.2) a psychological concern ( P = .711). Unicoronal: Mean ACFR with (0.65) and without (0.69) a psychological concern ( P = .423) were equivalent. However, mean ACFR with (0.74) and without (0.63) a speech-language concern were not ( P = .022*). Bivariate rank correlation showed significant association between morphologic severity and speech-language score only for unicoronal synostosis (ρ = .722; P = .008*). Conclusion: A significant portion of patients with SSC had speech-language or psychological concerns. We found no correlation between morphologic severity and incidence of speech-language or psychological concerns for patients with sagittal or metopic synostosis. Morphological severity did correlate with speech concerns in patients with unicoronal synostosis.

Comparison of Head Shape Outcomes in Metopic Synostosis Using Limited Strip Craniectomy and Open Vault Reconstruction Techniques

Aim: Metopic craniosynostosis (MCS), with its trigonocephalic head shape, is often treated with either limited incision strip craniectomy (LISC) followed by helmet orthotic treatment, or open cranial vault reconstruction techniques (OCVR). There is controversy regarding resultant shape outcomes among craniofacial surgeons. Those adverse to LISC claim normal head shape is never attained, while proponents believe there is gradual correction to an equivalent outcome. This study aims to quantitate, over time, the three-dimensional (3D) head shapes in patients who have undergone LISC or OCVR intervention for MCS. Methods: Sixty-three 3D images of 26 patients with MCS were analyzed retrospectively. Head shape analyses were performed at: (1) preoperative, (2) 1-month postoperative, (3) 10 to 14 months postoperative (1 year), and (4) 2 years postoperative. Composite 3D head shapes of patients were compared at each time point. Two-dimensional (2D) standardized cross sections of the forehead were also compared. Results: Composite head shapes for both groups were nested, to allow visual comparison as the child’s forehead grows and expands. The difference between LISC and OCVR 2D cross sections was calculated; 108.26 mm preoperatively, 127.18 mm after 1-month postoperative, 51.05 mm after 10 to 14 months postoperative, and 27.03 mm after 2 years postoperative. Conclusions: This study found excellent head shape outcomes for both the LISC and OCVR techniques at 2 years of age. It also corroborates the slow and progressive improvement in head shape with the LISC technique. This study highlights the advantages of 3D photography for measurement of contour outcomes, utilizing both 2D vector and 3D whole head analytical techniques.