Patient-reported outcome measures more than fifteen years after treatment of sagittal or metopic craniosynostosis: a prospective cohort study.

PURPOSE: To evaluate the long-term anthropometric measurements, cosmetic satisfaction, and other patient-reported outcome measures (PROMs) of patients who underwent surgical treatment or observation only of sagittal or metopic single-suture craniosynostosis (SSC). METHODS: A prospective study was designed for all patients diagnosed with non-syndromic sagittal and metopic craniosynostosis at the British Columbia Children's Hospital, Vancouver, Canada, in the period July 1986 to July 2006. After a minimum of 15 years post-diagnosis, all eligible patients were invited to fill out the Craniofacial Surgery Outcomes Questionnaire (CSO-Q) and to attend a scheduled follow-up appointment for the collection of anthropometric measurements. A descriptive analysis of the cosmetic results was performed. Statistical analyses compared the differences in anthropometric measurements between treated and non-treated patients. RESULTS: Of the 253 eligible patients, 52 participants were willing to share patient data for use in the study. Of those 52 former patients, 36 (69.2%) filled out and returned the CSO-Q and 23 (44.2%) attended the follow-up appointment. The mean follow-up period between surgical treatment and the CSO-Q was 20.2 ± 2.5 years and between surgical treatment and the follow-up appointment was 20.9 ± 2.7 years. In patients with sagittal SSC, the mean cephalic index (CI) was significantly larger in treated than in non-treated patients (74.6 versus 69.1, p = 0.04), while the mean pupillary distance and forehead to back index were significantly smaller (pupillary distance 6.0 cm versus 6.7 cm [p = 0.04] and forehead to back index 19.6 cm versus 21.1 cm [p = 0.03]). Focusing more on the patient reported outcome measures, overall cosmetic satisfaction was found to be high (80.6%) and no differences were found between sagittal and metopic synostosis patients, nor between treated or non-treated craniosynostosis patients. Overall outcomes regarding self-esteem (RSES) and fear of negative evaluation (FNE) were comparable with population based outcomes. CONCLUSION: This is the first prospective study of sagittal and metopic craniosynostosis patients regarding long-term anthropometric outcome and patient reported outcome measures, including patients who were treated surgically and those who received observation only. Although study participation two decades after initial diagnosis was difficult to obtain, our data provide a platform from which one can develop an inclusive and uniform approach to assess patients' subjective cosmetic satisfaction using the CSO-Questionnaire and might be useful in preoperative counseling and psychosocial care for patients and their families.

Performance of a diagnostic model for the presence of unruptured intracranial aneurysms in the general population.

INTRODUCTION: The prevalence of unruptured intracranial aneurysms (UIAs) in the general population is 3%. Aneurysmal subarachnoid hemorrhage (aSAH) can be prevented by screening for UIAs followed by monitoring and, if needed, preventive neurosurgical or endovascular treatment of identified UIAs. Therefore, we developed a diagnostic model for presence of UIAs in the general population to help identify persons at high risk of having UIAs. METHODS: Between 2005-2015, participants from the population-based Rotterdam Study underwent brain magnetic resonance imaging at 1.5 Tesla, on which presence of incidental UIAs was evaluated. We developed a multivariable logistic regression model using candidate diagnostic markers that were selected based on the literature, including sex, age, hypertension, smoking, hypercholesterolemia, diabetes, alcohol, and their interactions. We corrected for overfitting using bootstrapping. Model performance was assessed with discrimination, calibration, sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV). RESULTS: 5835 persons were included (55.0% women, mean age 64.9 ± 10.9 years) with a 2.2% UIA prevalence. Sex, age, hypertension, smoking, diabetes, and interactions of sex with age, hypertension, and smoking were independent diagnostic markers. The resulting model had a c-statistic of 0.65 (95% confidence interval [CI] 0.60 - 0.68) and 56% sensitivity, 52% specificity, 98% PPV, and 3% NPV for UIA presence at a cut-off value of 4%. Because of interactions with sex, additional models for men and women separately were developed. The model for men had a c-statistic of 0.70 (95% CI 0.62 - 0.78) with age, hypertension, and smoking as diagnostic markers and comparable additional performance values as for the full model. The model for women had a c-statistic of 0.58 (95% CI 0.52 - 0.63) with smoking as the only diagnostic marker. CONCLUSION: Our diagnostic model had insufficient performance to help identify persons at high risk of having UIAs in the general population. Rather, it provides insight in risk factors contributing to UIA risk and shows that these may be in part sex-specific.

Cosmetic satisfaction and patient-reported outcomes following surgical treatment of single-suture craniosynostosis: a systematic review.

PURPOSE: This study provides a systematic review on cosmetic satisfaction and other patient-reported outcomes (PROMs) of patients who underwent surgical treatment of SSC. METHODS: A systematic review of all articles published from inception to 1 June 2022 was performed. Articles were included if they reported on subjective assessment of cosmetic satisfaction or other PROMs by patients or their families using questionnaires or interviews. RESULTS: Twelve articles, describing 724 surgical treatments of SSC, met the inclusion criteria. Cosmetic satisfaction was evaluated in the following ways: 1) use of the VAS score, binary questions or a 5-point scale to rate general, facial or skull appearance; 2) use of an aesthetic outcome staging in which personal opinion was added to the treating surgeon's opinion; and 3) use of an evaluation of anatomical proportions of the skull and face. A trend towards an overall improvement in cosmetic satisfaction following surgical treatment of SSC was observed. Reported PROMs included general health, socioeconomic status, patients' and their families' rating of the normalcy and noticeability of their appearance and how much this bothered them, and patients' answers to the Youth Quality of Life with Facial Differences (YQOL-FD) questionnaire. No clear overall trend of the reported PROMs was identified. CONCLUSION: This systematic review illuminates that there is a wide variation in outcomes for evaluating cosmetic satisfaction and other PROMs of patients who underwent surgical treatment of SSC, suggesting that further research is needed to develop an inclusive and uniform approach to assess these outcomes.

Cosmetic satisfaction and patient-reported outcome measures following cranioplasty after craniectomy - A prospective cohort study.

Introduction: Evaluating patient-reported outcomes (PROMs) helps optimize preoperative counseling and psychosocial care for patients who underwent cranioplasty. Research question: This study aimed to evaluate cosmetic satisfaction, level of self-esteem, and fear of negative evaluation (FNE) of patients who underwent cranioplasty. Material and methods: Patients who underwent cranioplasty from 1 January 2014 to 31 December 2020 â€‹at University Medical Center Utrecht and a control group consisting of our center' employees were invited to fill out the Craniofacial Surgery Outcomes Questionnaire (CSO-Q), consisting of an assessment of cosmetic satisfaction, the Rosenberg Self-Esteem Scale (RSES), and the FNE scale. To test for differences in results, chi-square tests and T-tests were performed. Logistic regression was used to study the effect of cranioplasty-related variables on cosmetic satisfaction. Results: Cosmetic satisfaction was seen in 44/80 patients (55.0%) and 52/70 controls (74.3%) (p â€‹= â€‹0.247). Thirteen patients (16.3%) and 8 controls (11.4%) had high self-esteem (p â€‹= â€‹0.362), 51 patients (63.8%) and 59 controls (84.3%) had normal self-esteem (p â€‹= â€‹0.114), and 7 patients (8.8%) and 3 controls (4.3%) had low self-esteem (p â€‹= â€‹0.337). Forty-nine patients (61.3%) and 39 controls (55.7%) had low FNE (p â€‹= â€‹0.012), 8 patients (10.0%) and 18 controls (25.7%) had average FNE (p â€‹= â€‹0.095), and 6 patients (7.5%) and 13 controls (18.6%) had high FNE (p â€‹= â€‹0.215). Cosmetic satisfaction was associated with glass fiber-reinforced composite implants (OR 8.20, p-value â€‹= â€‹0.04). Discussion and conclusion: This study prospectively evaluated PROMs following cranioplasty, for which we found favorable results.

Development of a Prediction Model for Cranioplasty Implant Survival Following Craniectomy.

BACKGROUND: Cranioplasty after craniectomy can result in high rates of postoperative complications. Although determinants of postoperative outcomes have been identified, a prediction model for predicting cranioplasty implant survival does not exist. Thus, we sought to develop a prediction model for cranioplasty implant survival after craniectomy. METHODS: We performed a retrospective cohort study of patients who underwent cranioplasty following craniectomy between 2014 and 2020. Missing data were imputed using multiple imputation. For model development, multivariable Cox proportional hazards regression analysis was performed. To test whether candidate determinants contributed to the model, we performed backward selection using the Akaike information criterion. We corrected for overfitting using bootstrapping techniques. The performance of the model was assessed using discrimination and calibration. RESULTS: A total of 182 patients were included (mean age, 43.0 ± 19.7 years). Independent determinants of cranioplasty implant survival included the indication for craniectomy (compared with trauma-vascular disease: hazard ratio [HR], 0.65 [95% confidence interval (CI), 0.36-1.17]; infection: HR, 0.76 [95% CI, 0.32-1.80]; tumor: HR, 1.40 [95% CI, 0.29-6.79]), cranial defect size (HR, 1.01 per cm2 [95% CI, 0.73-1.38]), use of an autologous bone flap (HR, 1.63 [95% CI, 0.82-3.24]), and skin closure using staples (HR, 1.42 [95% CI, 0.79-2.56]). The concordance index of the model was 0.60 (95% CI, 0.47-0.73). CONCLUSIONS: We have developed the first prediction model for cranioplasty implant survival after craniectomy. The findings from our study require external validation and deserve further exploration in future studies.

Cranial decompression and expansion surgery for the treatment of refractory idiopathic intracranial hypertension: case report and systematic review.

OBJECTIVE: The purpose of this study is to systematically review the literature on the clinical outcomes following different surgical techniques in patients with refractory idiopathic intracranial hypertension (IIH). BACKGROUND: IIH is a condition characterised by increased cranial pressure (ICP) in the absence of an intracranial lesion that does not adequately respond to different medical and surgical therapies. Cranial decompression or expansion surgeries are a last resort therapy for patients with refractory IIH. METHODS: A systematic literature search of the databases of PubMed, Embase and Medline from inception to 2019 was performed. Searches were limited to the English language and to clinical studies. Studies were included if clinical outcomes following different cranial decompression or expansion techniques were reported. We also add one case of our own experience with performing a bilateral frontoparietal expansion craniotomy and subtemporal craniectomy. RESULTS: Five manuscripts, describing 38 procedures, met the inclusion criteria. Thirty-one patients were female (82%). The mean age was 26.2 years. The techniques studied included subtemporal craniectomy (27/38, 71%), internal cranial expansion (10/38, 26%), and cranial morcellation decompression (1/38, 3%). Thirty-five patients presented with headaches of which 17 noted postoperative improvement or resolution (49%). Visual deficits were documented in 30 patients and 25 reported postoperative improvement (83%). Papilledema disappeared in 23 of 32 patients with this sign at presentation (72%). In our patient, symptoms completely resolved postoperatively and a 6% increase in intracranial volume was measured. CONCLUSIONS: Cranial vault decompression or expansion surgeries may be an effective last resort therapy for patients with refractory IIH. These surgeries expand the intracranial volume, and thus may normalise ICP, leading to clinical improvement.

Cranioplasty after craniectomy in pediatric patients-a systematic review.

INTRODUCTION: Complications following cranioplasty with either autografts or cranial implants are commonly reported in pediatric patients. However, data regarding cranioplasty strategies, complications and long-term outcomes are not well described. This study systematically reviews the literature for an overview of current cranioplasty practice in children. METHODS: A systematic review of articles published from inception to July 2018 was performed. Studies were included if they reported the specific use of cranioplasty materials following craniectomy in patients younger than 18 years of age, and had a minimum follow-up of at least 1 year. RESULTS: Twenty-four manuscripts, describing a total of 864 cranioplasty procedures, met the inclusion criteria. The age of patients in this aggregate ranged from 1 month to 20 years and the weighted average was 8.0 years. The follow-up ranged from 0.4 months to 18 years and had a weighted average of 40.4 months. Autologous bone grafts were used in 484 cases (56.0%). Resorption, infection and/or hydrocephalus were the most frequently mentioned complications. In this aggregate group, 61 patients needed a revision cranioplasty. However, in 6/13 (46%) papers studying autologous cranioplasties, no data was provided on resorption, infection and revision cranioplasty rates. Cranial implants were used in 380 cases (44.0%), with custom-made porous hydroxyapatite being the most commonly used material (100/380, 26.3%). Infection and migration/fracturing/loosening were the most frequently documented complications. Eleven revision cranioplasties were reported. Again, no data was reported on infection and revision cranioplasty rates, in 7/16 (44%) and 9/16 (56%) of papers, respectively. CONCLUSION: Our systematic review illuminates that whether autografts or cranial implants are used, postcranioplasty complications are quite common. Beyond this, the existing literature does not contain well documented and comparable outcome parameters, suggesting that prospective, long-term multicenter cohort studies are needed to be able to optimize cranioplasty strategies in children who will undergo cranioplasty following craniectomy.

Cranioplasties following craniectomies in children-a multicenter, retrospective cohort study.

OBJECTIVE: Complications following pediatric cranioplasty after craniectomy with either autologous bone flaps or cranial implants are reported to be common, particularly bone flap resorption. However, only sparse data are available regarding cranioplasty strategies, complications, and outcomes. This manuscript describes a Canadian-Dutch multicenter pediatric cohort study with autografts and cranial implant cranioplasties following craniectomies for a variety of indications. METHODS: The study included all children (< 18 years) who underwent craniectomy and subsequent cranioplasty surgeries from 2008 to 2014 (with a minimum of 1-year follow-up) at four academic hospitals with a dedicated pediatric neurosurgical service. Data were collected regarding initial diagnosis, age, time interval between craniectomy and cranioplasty, bone flap storage method, type of cranioplasty for initial procedure (and redo if applicable), and the postoperative outcome including surgical site infection, wound breakdowns, bone flap resorption, and inadequate fit/disfigurement. RESULTS: Sixty-four patients (46 males, average age 9.7 ± 5.5 years) were eligible for inclusion, with mean follow-up of 82.3 ± 31.2 months after craniectomy. Forty cranioplasties (62.5%) used autologous bone re-implant, 23 (57.5%) of which showed resorption. On average, resorption was documented at 434 days (range 62-2796 days) after reimplantation. In 20 cases, a revision cranioplasty was needed. In 24 of the post-craniectomy cases (37.5%), a cranial implant was used with one of ten different implant types. Implant loosening prompted a complete revision cranioplasty in 2 cases (8.3%). Cranial implants were associated with low morbidity and lower reoperation dates compared to the autologous cranioplasties. CONCLUSION: The most prominent finding in this multicenter cohort study was that bone flap resorption in children remains a common and widespread problem following craniectomy. Cranioplasty strategies varied between centers and evolved over time within centers. Cranial implants were associated with low morbidity and low reoperation rates. Still, longer term and prospective multicenter cohort studies are needed to optimize cranioplasty strategies in children after craniectomies.