To identify skull-base growth patterns in Crouzon syndrome, we hypothesized premature minor suture fusion restricts occipital bone development, secondarily limiting foramen magnum expansion. Skull-base suture closure degree and cephalometric measurements were retrospectively studied using preoperative computed tomography (CT) scans and multiple linear regression analysis. Evaluation of multi-institutional CT images and 3D reconstructions from Wake Forest’s Craniofacial Imaging Database (WFCID). Sixty preoperative patients with Crouzon syndrome under 12 years-old were selected from WFCID. The control group included 60 age- and sex-matched patients without craniosynostosis or prior craniofacial surgery. None 2D and 3D cephalometric measurements. 3D volumetric evaluation of the basioccipital, exo-occipital, and supraoccipital bones revealed decreased growth in Crouzon syndrome, attributed solely to premature minor suture fusion. Spheno-occipital (β = −398.75; P < .05) and petrous-occipital (β = −727.5; P < .001) suture fusion reduced growth of the basioccipital bone; lambdoid suture (β = −14 723.1; P < .001) and occipitomastoid synchondrosis (β = −16 419.3; P < .001) fusion reduced growth of the supraoccipital bone; and petrous-occipital suture (β = −673.3; P < .001), anterior intraoccipital synchondrosis (β = −368.47; P < .05), and posterior intraoccipital synchondrosis (β = −6261.42; P < .01) fusion reduced growth of the exo-occipital bone. Foramen magnum morphology is restricted in Crouzon syndrome but not directly caused by early suture fusion. Premature minor suture fusion restricts the volume of developing occipital bones providing a plausible mechanism for observed foramen magnum anomalies.
Background: Frontal bossing is a prominent forehead feature common in sagittal craniosynostosis (SC). Assessment of severity of frontal bossing is limited by the use of serial CT imaging or complex computer programing. Three-dimensional measurements of cranial surface morphology provide a radiation-free alternative for assessing cranial shape. This study describes the creation of a frontal bossing index (FBI), a novel tool using surface morphology to assess the frontal severity in patients with SC. Methods: Surface imaging from CT scans or 3D photographs of 359 individuals with sagittal craniosynostosis and 224 normocephalic individuals were compared to identify differences in frontal morphology. Cartesian grids were created on each individual’s surface mesh using equidistant axial and sagittal planes yielding 33 unique points of intersection on the forehead. Area under the curve (AUC) analyses were performed to identify frontal points with the greatest discrepancy between groups. Results: All points in the SC population had significantly greater protrusion than corresponding points in the control group. The largest differences were located in the superior lateral regions. Combining the superior- and lateral-most points gave the maximal AUC (0.9707) and was therefore selected to generate the frontal bossing index (FBI). The FBI distinguished between the 2 groups with a sensitivity of 93.5% and specificity of 92.9%. Conclusions: The frontal bossing index is a useful tool for evaluating the severity of the frontal region in patients with SC, comparing outcomes of differing surgical techniques, and tracking frontal changes in individuals over time, without the need for radiation.
Background: There is debate on the utility of a preoperative Allen test or ultrasound before radial forearm free flap (RFFF) harvest. This study sought to evaluate correlations between preoperative testing and donor-site morbidity. Methods: A survey of plastic surgery and otolaryngology RFFF patients was conducted at a Midwestern academic center. The modified Cold Intolerance Symptom Severity (modCISS) and Quick Disabilities of the Arm, Shoulder, and Hand (QDASH) instruments were administered. A retrospective chart review was performed to assess perioperative factors. Results: Of 212 RFFFs completed over 7 years, 144 patients were contacted, and 71 patients completed the survey (33% response rate). Preoperative Allen test was negative in 92% of patients (65 of 71). There was no statistical association between Allen test and duplex ultrasound findings (P = 0.19). Cold intolerance screening was positive on 20% of donor arms (14 of 71), with an average positive modCISS score of 39.0 ± 14.7. Disability was reported on the QDASH by 76% of patients (54 of 71), with an average score of 21.0 ± 22.3. There was no statistical correlation between preoperative Allen test or ultrasound classification and modCISS or QDASH score. There was a borderline positive correlation between modCISS and QDASH scores that did not reach statistical significance (r = 0.22, P = 0.067). Operative characteristics also did not predict modCISS or QDASH scores. Conclusions: Following RFFF harvest, donor extremity cold intolerance is reported in 20% of patients, and extremity-related disability is reported in the majority of patients. Preoperative ultrasound and physical examination findings are not predictive of morbidity.
Background: Endoscopically assisted craniofacial surgery (EACS) has numerous advantages over traditional, open approaches, such as fronto-orbital advancement in treating nonsyndromic craniosynostosis. However, several articles report high reoperation rates in syndromic patients treated with EACS. This meta-analysis and review examines undesirable outcome rates (UORs), defined as reoperation or Whitaker category III/IV, in syndromic patients undergoing primary EACS compared with procedures that actively expand the cranial vault. Methods: PubMed and Embase were searched in June 2022 to identify all articles reporting primary reoperation or Whitaker outcomes for syndromic patients undergoing cranial vault expanding surgery or suturectomy. A meta-analysis of proportions was performed comparing UORs, and a trim-and-fill adjustment method was used to validate sensitivity and assess publication bias. Results: A total of 721 articles were screened. Five EACS articles (83 patients) and 22 active approach articles (478 patients) met inclusion criteria. Average UORs for EACS and active approaches were 26% (14%–38%) and 20% (13%–28%), respectively (P = 0.18). Reoperation occurred earlier in EACS patients (13.7 months postprimary surgery versus 37.1 months for active approaches, P = 0.003). Relapse presentations and reason for reoperation were also reviewed. Subjectively, EACS UORs were higher in all syndromes except Apert, and Saethre-Chotzen patients had the highest UOR for both approaches. Conclusions: There was no statistically significant increase in UORs among syndromic patients treated with EACS compared with traditional approaches, although EACS patients required revision significantly sooner. Uncertainties regarding the long-term efficacy of EACS in children with syndromic craniosynostosis should be revisited as more data become available.
Background: Sagittal craniosynostosis (SC) is associated with scaphocephaly, an elongated narrow head shape. Assessment of regional severity in the scaphocephalic head is limited by the use of serial CT imaging or complex computer programing. Three-dimensional measurements of cranial surface morphology provide a radiation-free alternative for assessing cranial shape. This study describes the creation of an Occipital Bulleting Index (OBI), a novel tool using surface morphology to assess the regional severity in patients with SC. Methods: Surface imaging from CT scans or 3D photographs of 360 individuals with sagittal craniosynostosis and 221 normocephalic individuals were compared to identify differences in morphology. Cartesian grids were created on each individual’s surface mesh using equidistant axial and sagittal planes. Area under the curve (AUC) analyses were performed to identify trends in regional morphology and create measures capturing population differences.Results: The largest differences were located in the medial regions posteriorly. Using these population trends, a measure was created maximizing AUC. The Occipital Bullet index has an AUC of 0.72 with a sensitivity of 74% and a specificity of 61%. When the Frontal Bossing Index is applied in tandem, the two have a sensitivity of 94.7% and a specificity of 93.1%. Correlation between the two scores in individuals with SC was found to be negligible with an intraclass correlation coefficient of 0.018. Severity was found to be independent of age under 24 months, sex, and imaging modality.Conclusions: This index creates a tool for differentiating control head shapes from those with sagittal craniosynostosis, and has the potential to allow for objective evaluation of the regional severity, outcomes of different surgical techniques, and tracking shape changes in individuals over time, without the need for radiation.
Purpose: Early onset of minor suture fusion in syndromic craniosynostosis is associated with midface dysplasia and is a common indication for craniofacial surgery. However, the potential effects of fusion severity on craniofacial growth patterns are not well understood. This study seeks to describe the impact of minor suture fusion severity on midface morphology in Crouzon syndrome. Methods: Pre-operative computed tomography images (CT) of 63 patients with Crouzon syndrome and 63 normocepahlic controls were included. Degree of suture fusion was assessed for the frontosphenoidal, sphenethmoidal, sphenosquamosal, sphenopetrosal, spheno-occipital synchondrosis, frontoethmoidal, and zygomaticosphenoidal sutures. Each suture was graded on a 5-point scale. The sella (S), nasion (N), A point (A), basion (BA), and anterior nasal spine (ANS) landmarks were used to calculate the SNA angle, BA-ANS length of the lower midface, and N-S length of the upper midface. Multiple linear regressions were used to analyze data. Results: The mean age was 43 months and 44% were female. The control group was significantly older ( P < .01) than the patients with Crouzon syndrome. Advanced fusion of the spheno-occipital synchondrosis in Crouzon syndrome correlates with regression of the BA-ANS length by 0.563 mm per incremental increase in suture fusion ( P < .01). Additionally, the lower midface (BA-ANS) was restricted to a greater degree than the upper midface (N-S) with progressive suture fusion in all patient types with ratios of these rates ranging between 0.602 and 0.89 for the 7 sutures analyzed. Suture fusion severity did not impact the SNA angle in any of the analyses performed. Conclusion: The severity of sheno-occipital synchondrosis fusion in Crouzon syndrome contributes to midface hypoplasia. Similarly, all anterior skull base sutures limited lower midface growth to a greater degree than the upper midface.
CONCLUSION: Neighborhood measures of disadvantage impact interventions, complications, and outcomes in patients with craniosynostosis and patients with cleft palate. Further inquiry into the extent to which socioeconomic situation contributes to these risks can inform interventions to optimize treatment and outcomes in both patient populations.
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