ObjectiveValidation of automated 2-dimensional (2D) diameter measurements of vestibular schwannomas on magnetic resonance imaging (MRI). Study DesignRetrospective validation study using 2 data sets... Show moreObjectiveValidation of automated 2-dimensional (2D) diameter measurements of vestibular schwannomas on magnetic resonance imaging (MRI). Study DesignRetrospective validation study using 2 data sets containing MRIs of vestibular schwannoma patients. SettingUniversity Hospital in The Netherlands. MethodsTwo data sets were used, 1 containing 1 scan per patient (n = 134) and the other containing at least 3 consecutive MRIs of 51 patients, all with contrast-enhanced T1 or high-resolution T2 sequences. 2D measurements of the maximal extrameatal diameters in the axial plane were automatically derived from a 3D-convolutional neural network compared to manual measurements by 2 human observers. Intra- and interobserver variabilities were calculated using the intraclass correlation coefficient (ICC), agreement on tumor progression using Cohen's kappa. ResultsThe human intra- and interobserver variability showed a high correlation (ICC: 0.98-0.99) and limits of agreement of 1.7 to 2.1 mm. Comparing the automated to human measurements resulted in ICC of 0.98 (95% confidence interval [CI]: 0.974; 0.987) and 0.97 (95% CI: 0.968; 0.984), with limits of agreement of 2.2 and 2.1 mm for diameters parallel and perpendicular to the posterior side of the temporal bone, respectively. There was satisfactory agreement on tumor progression between automated measurements and human observers (Cohen's & kappa; = 0.77), better than the agreement between the human observers (Cohen's & kappa; = 0.74). ConclusionAutomated 2D diameter measurements and growth detection of vestibular schwannomas are at least as accurate as human 2D measurements. In clinical practice, measurements of the maximal extrameatal tumor (2D) diameters of vestibular schwannomas provide important complementary information to total tumor volume (3D) measurements. Combining both in an automated measurement algorithm facilitates clinical adoption. Show less
Objective: To analyze the effect of dizziness-related symptoms on the long-term quality of life (QoL) of patients with unilateral vestibular schwannoma. Methods: In this cross-sectional study,... Show moreObjective: To analyze the effect of dizziness-related symptoms on the long-term quality of life (QoL) of patients with unilateral vestibular schwannoma. Methods: In this cross-sectional study, patients with a unilateral vestibular schwannoma diagnosed between 2004 and 2013 completed a disease-specific QoL questionnaire (Penn Acoustic Neuroma Quality of Life [PANQOL]) and the Dizziness Handicap Inventory (DHI) in 2020. Linear regression was performed to assess the correlation between QoL and the DHI total score, and the scores of the DHI functional, emotional, and physical subdomains. Potential confounders such as age, sex, tumor size at baseline, and treatment modality (active surveillance, surgery, or radiotherapy) were included in the model. Results: In total, 287 of 479 patients (59%) experienced dizziness with a median follow-up of 10 years. The DHI total score was significantly associated with the PANQOL total score. On average, we found a reduction of 0.7 points on the PANQOL for each additional point on the DHI. The DHI emotional subdomain was the most prominent determinant of poorer QoL. Each point on the DHI emotional subscale was associated with a reduction of 1.3 on the PANQOL score. Treatment modality did not have a clinically relevant effect on dizziness-related QoL. Conclusions: Even mild dizziness can have a significant and clinically relevant effect on the QoL of patients with unilateral vestibular schwannoma in the long term. This holds true for all treatment modalities. Addressing the vestibular problems may improve QoL in vestibular schwannoma patients, and DHI subscale analysis may help tailor the optimal vestibular intervention. Show less
Background Evaluating patients' experiences is essential when incorporating the patients' perspective in improving healthcare. Experiences are mainly collected using closed-ended questions,... Show moreBackground Evaluating patients' experiences is essential when incorporating the patients' perspective in improving healthcare. Experiences are mainly collected using closed-ended questions, although the value of open-ended questions is widely recognized. Natural language processing (NLP) can automate the analysis of open-ended questions for an efficient approach to patient-centeredness. Methods We developed the Artificial Intelligence Patient-Reported Experience Measures (AI-PREM) tool, consisting of a new, open-ended questionnaire, an NLP pipeline to analyze the answers using sentiment analysis and topic modeling, and a visualization to guide physicians through the results. The questionnaire and NLP pipeline were iteratively developed and validated in a clinical context. Results The final AI-PREM consisted of five open-ended questions about the provided information, personal approach, collaboration between healthcare professionals, organization of care, and other experiences. The AI-PREM was sent to 867 vestibular schwannoma patients, 534 of which responded. The sentiment analysis model attained an F1 score of 0.97 for positive texts and 0.63 for negative texts. There was a 90% overlap between automatically and manually extracted topics. The visualization was hierarchically structured into three stages: the sentiment per question, the topics per sentiment and question, and the original patient responses per topic. Conclusions The AI-PREM tool is a comprehensive method that combines a validated, open-ended questionnaire with a well-performing NLP pipeline and visualization. Thematically organizing and quantifying patient feedback reduces the time invested by healthcare professionals to evaluate and prioritize patient experiences without being confined to the limited answer options of closed-ended questions. Show less
Neve, O.M.; Chen, Y.J.; Tao, Q.; Romeijn, S.R.; Boer, N.P. de; Grootjans, W.; ... ; Staring, M. 2022
Purpose: To develop automated vestibular schwannoma measurements on contrast-enhanced T1- and T2-weighted MRI scans.Materials and Methods: MRI data from 214 patients in 37 different centers were... Show morePurpose: To develop automated vestibular schwannoma measurements on contrast-enhanced T1- and T2-weighted MRI scans.Materials and Methods: MRI data from 214 patients in 37 different centers were retrospectively analyzed between 2020 and 2021. Patients with hearing loss (134 positive for vestibular schwannoma [mean age 6 SD, 54 years 6 12; 64 men] and 80 negative for vestibular schwannoma) were randomly assigned to a training and validation set and to an independent test set. A convolutional neural network (CNN) was trained using fivefold cross-validation for two models (T1 and T2). Quantitative analysis, including Dice index, Hausdorff distance, surface-to-surface distance (S2S), and relative volume error, was used to compare the computer and the human delineations. An observer study was performed in which two experienced physicians evaluated both delineations.Results: The T1-weighted model showed state-of-the-art performance, with a mean S2S distance of less than 0.6 mm for the whole tumor and the intrameatal and extrameatal tumor parts. The whole tumor Dice index and Hausdorff distance were 0.92 and 2.1 mm in the independent test set, respectively. T2-weighted images had a mean S2S distance less than 0.6 mm for the whole tumor and the intrameatal and extrameatal tumor parts. The whole tumor Dice index and Hausdorff distance were 0.87 and 1.5 mm in the independent test set. The observer study indicated that the tool was similar to human delineations in 85%-92% of cases.Conclusion: The CNN model detected and delineated vestibular schwannomas accurately on contrast-enhanced T1- and T2-weighted MRI scans and distinguished the clinically relevant difference between intrameatal and extrameatal tumor parts. (C) RSNA, 2022 Show less
Neve, O.M.; Jansen, J.C.; Koot, R.W.; Ridder, M. de; Benthem, P.P.G. van; Stiggelbout, A.M.; Hensen, E.F. 2022
Objective: Vestibular schwannoma management aims to maintain optimal quality of life (QoL) while preventing severe sequelae of the tumor or its treatment. This study assessed long-term QoL of... Show moreObjective: Vestibular schwannoma management aims to maintain optimal quality of life (QoL) while preventing severe sequelae of the tumor or its treatment. This study assessed long-term QoL of patients with vestibular schwannoma in relation to treatment modality and decisional regret. Study Design: A longitudinal study, in which clinical and QoL data were used that were cross-sectionally acquired in 2014 and again in 2020 from the same patient group. Setting: A tertiary expert center for vestibular schwannoma care in the Netherlands. Methods: QoL was measured by the Penn Acoustic Quality of Life (PANQOL) scale. Changes in time were assed using a linear mixed model. In addition, the Decision Regret Scale was analyzed. Results: Of 867 patients, 536 responded (62%), with a median follow-up of 11 years. All PANQOL subdomain scores remained stable over time and did not exceed minimal clinically important difference (MCID) levels. Time since treatment did not affect QoL. Patients had comparable average QoL scores and proportions of patients with changing QoL scores (ie, exceeding the MCID) over time, irrespective of the received initial treatment. Female patients and those who required salvage therapy (either by radiotherapy or surgery) reported a lower QoL. The latter patient group reported the highest decisional regret. Conclusion: On average, the long-term QoL of patients with vestibular schwannoma is comparable for patients under active surveillance and those who have received active treatment, and it remains stable over time. This suggests that, on average, preservation of QoL of patients with vestibular schwannoma is feasible when adequately managed. Show less
Neve, O.M.; Jansen, J.C.; Koot, R.W.; Ridder, M. de; Benthem, P.P.G. van; Stiggelbout, A.M.; Hensen, E.F. 2022
Objective Vestibular schwannoma management aims to maintain optimal quality of life (QoL) while preventing severe sequelae of the tumor or its treatment. This study assessed long-term QoL of... Show moreObjective Vestibular schwannoma management aims to maintain optimal quality of life (QoL) while preventing severe sequelae of the tumor or its treatment. This study assessed long-term QoL of patients with vestibular schwannoma in relation to treatment modality and decisional regret. Study Design A longitudinal study, in which clinical and QoL data were used that were cross-sectionally acquired in 2014 and again in 2020 from the same patient group. Setting A tertiary expert center for vestibular schwannoma care in the Netherlands. Methods QoL was measured by the Penn Acoustic Quality of Life (PANQOL) scale. Changes in time were assed using a linear mixed model. In addition, the Decision Regret Scale was analyzed. Results Of 867 patients, 536 responded (62%), with a median follow-up of 11 years. All PANQOL subdomain scores remained stable over time and did not exceed minimal clinically important difference (MCID) levels. Time since treatment did not affect QoL. Patients had comparable average QoL scores and proportions of patients with changing QoL scores (ie, exceeding the MCID) over time, irrespective of the received initial treatment. Female patients and those who required salvage therapy (either by radiotherapy or surgery) reported a lower QoL. The latter patient group reported the highest decisional regret. Conclusion On average, the long-term QoL of patients with vestibular schwannoma is comparable for patients under active surveillance and those who have received active treatment, and it remains stable over time. This suggests that, on average, preservation of QoL of patients with vestibular schwannoma is feasible when adequately managed. Show less
Neve, O.M.; Benthem, P.P.G. van; Stiggelbout, A.M.; Hensen, E.F. 2021
Background Patient Reported Outcomes (PROs) are subjective outcomes of disease and/or treatment in clinical research. For effective evaluations of PROs, high response rates are crucial. This study... Show moreBackground Patient Reported Outcomes (PROs) are subjective outcomes of disease and/or treatment in clinical research. For effective evaluations of PROs, high response rates are crucial. This study assessed the impact of the delivery method on the patients' response rate. Methods A cohort of patients with a unilateral vestibular schwannoma (a condition with substantial impact on quality of life, requiring prolonged follow-up) was assigned to three delivery methods: email, regular mail, and hybrid. Patients were matched for age and time since the last visit to the outpatient clinic. The primary outcome was the response rate, determinants other than delivery mode were age, education and time since the last consultation. In addition, the effect of a second reminder by telephone was evaluated. Results In total 602 patients participated in this study. The response rates for delivery by email, hybrid, and mail were 45, 58 and 60%, respectively. The response rates increased after a reminder by telephone to 62, 67 and 64%, respectively. A lower response rate was associated with lower level of education and longer time interval since last outpatient clinic visit. Conclusion The response rate for PRO varies by delivery method. PRO surveys by regular mail yield the highest response rate, followed by hybrid and email delivery methods. Hybrid delivery combines good response rates with the ease of digitally returned questionnaires. Show less
Neve, O.M.; Boerman, J.A.; Hout, W.B. van den; Briaire, J.J.; Benthem, P.P.G. van; Frijns, J.H.M. 2021
Objectives: While the costs and outcomes of cochlear implantation (CI) have been widely assessed, most of these analyses were solely performed from the perspective of healthcare costs. This study... Show moreObjectives: While the costs and outcomes of cochlear implantation (CI) have been widely assessed, most of these analyses were solely performed from the perspective of healthcare costs. This study assesses the costs and benefits of CI in the Netherlands from a broader societal perspective, including health outcomes, healthcare cost, educational cost, and productivity losses and gains. Design: The cost and benefits of CI were analyzed in this cost-benefit analysis, in which a monetary value is put on both the resources needed and the outcomes of CI. The costs and benefits were analyzed by prototypical instances of three groups, representing the majority of cochlear implant patients: prelingually deaf children implanted at the age of 1, adults with progressive profound hearing loss implanted at the age of 40 and seniors implanted at the age of 70 with progressive profound hearing loss. Costs and benefits were estimated over the expected lifetimes of the members of each group, using a Markov state transition model. Model parameters and assumptions were based on published literature. Probabilistic and one-way sensitivity analyses were performed. Results: In all three patient groups, the total benefits of CI exceeded the total cost, leading to a net benefit of CI. Prelingually deaf children with a bilateral CI had a lifetime positive outcome net benefit of euro433,000. Adults and seniors with progressive profound hearing loss and a unilateral CI had a total net benefit of euro275,000 and euro76,000, respectively. These results ensue from health outcomes expressed in monetary terms, reduced educational cost, and increased productivity. Conclusions: Based on estimates from modeling, the increased healthcare costs due to CI were more than compensated by the value of the health benefits and by savings in educational and productivity costs. In particular, for children and working adults, the societal benefit was positive even without taking health benefits into account. Therefore, CI generates an advantage for both patients and society. Show less
Neve, O.M.; Jansen, J.C.; Mey, A.G.L. van der; Koot, R.W.; Ridder, M. de; Benthem, P.P.G. van; ... ; Hensen, E.F. 2021
Background Employment is an important factor in quality of life. For vestibular schwannoma (VS) patients, employment is not self-evident, because of the sequelae of the disease or its treatment and... Show moreBackground Employment is an important factor in quality of life. For vestibular schwannoma (VS) patients, employment is not self-evident, because of the sequelae of the disease or its treatment and their effects on daily life. Objectives This study assessed employment status, sick leave (absenteeism) and being less productive at work (presenteeism) in the long-term follow-up of VS patients, and evaluated the impact of treatment strategy (active surveillance, surgery or radiotherapy). Methods A cross-sectional survey study was performed in a tertiary university hospital in the Netherlands. Patients completed the iMTA-post productivity questionnaire (iPCQ). Employment status was compared to that of the general Dutch population. Employment, absenteeism and presenteeism were compared between patients under active surveillance, patients after radiotherapy and post-surgical patients. Result In total 239 patients participated, of which 67% were employed at the time of the study. Only 14% had a disability pension, which was comparable to the age-matched general Dutch population. The proportion of patients with absenteeism was 8%, resulting in a 4% reduction of working hours. Presenteeism was reported by 14% of patients, resulting in a 2% reduction of working hours. The median number of working hours per week was 36, and since the diagnosis, these hours had been reduced by 6%. There were no significant differences between treatment modalities. Conclusion On average, long-term employment status and working hours of VS patients are comparable to the age-matched general population. Treatment strategies do not seem to differentially impact on long-term employment of VS patients. Show less
Neve, O.M.; Soulier, G.; Hendriksma, M.; Mey, A.G.L. van der; Linge, A. van; Benthem, P.P.G. van; ... ; Stiggelbout, A.M. 2020
Purpose In cases of small- to medium-sized vestibular schwannomas, three management strategies can be opted for: active surveillance, surgery or radiotherapy. In these cases, the patient's... Show morePurpose In cases of small- to medium-sized vestibular schwannomas, three management strategies can be opted for: active surveillance, surgery or radiotherapy. In these cases, the patient's preference is pivotal in decision-making. The aim of this study was to identify factors that influence a patient's decision for a particular management strategy. Methods A qualitative inductive thematic analysis was performed based on semi-structured interviews. Eighteen patients with small- to medium-sized vestibular schwannomas were interviewed. All patients were diagnosed or treated at one of the two participating university medical centers in the Netherlands. Results Ten themes were identified that influenced the decision, classified as either medical or patient-related. The medical themes that emerged were: tumor characteristics, the physician's recommendation, treatment outcomes and the perceived center's experience. The patient-related themes were: personal characteristics, anxiety, experiences, cognitions, logistics and trust in the physician. Conclusion Knowledge of the factors that influence decision-making helps physicians to tailor their consultations to arrive at a true shared decision on vestibular schwannoma management. Show less
Neve, O.M.; Soulier, G.; Hendriksma, M.; Mey, A.G.L. van der; Linge, A. van; Benthem, P.P.G. van; ... ; Stiggelbout, A.M. 2020
AbstractPurposeIn cases of small- to medium-sized vestibular schwannomas, three management strategies can be opted for: active surveillance, surgery or radiotherapy. In these cases, the patient’s... Show moreAbstractPurposeIn cases of small- to medium-sized vestibular schwannomas, three management strategies can be opted for: active surveillance, surgery or radiotherapy. In these cases, the patient’s preference is pivotal in decision-making. The aim of this study was to identify factors that influence a patient’s decision for a particular management strategy.MethodsA qualitative inductive thematic analysis was performed based on semi-structured interviews. Eighteen patients with small- to medium-sized vestibular schwannomas were interviewed. All patients were diagnosed or treated at one of the two participating university medical centers in the Netherlands.ResultsTen themes were identified that influenced the decision, classified as either medical or patient-related. The medical themes that emerged were: tumor characteristics, the physician’s recommendation, treatment outcomes and the perceived center’s experience. The patient-related themes were: personal characteristics, anxiety, experiences, cognitions, logistics and trust in the physician.ConclusionKnowledge of the factors that influence decision-making helps physicians to tailor their consultations to arrive at a true shared decision on vestibular schwannoma management. Show less
