The Development, Deployment, and Evaluation of the CLEFT-Q Computerized Adaptive Test:A Multimethods Approach Contributing to Personalized, Person-Centered Health Assessments in Plastic Surgery

BackgroundRoutine use of patient-reported outcome measures (PROMs) and computerized adaptive tests (CATs) may improve care in a range of surgical conditions. However, most available CATs are neither condition-specific nor coproduced with patients and lack clinically relevant score interpretation. Recently, a PROM called the CLEFT-Q has been developed for use in the treatment of cleft lip or palate (CL/P), but the assessment burden may be limiting its uptake into clinical practice. ObjectiveWe aimed to develop a CAT for the CLEFT-Q, which could facilitate the uptake of the CLEFT-Q PROM internationally. We aimed to conduct this work with a novel patient-centered approach and make source code available as an open-source framework for CAT development in other surgical conditions. MethodsCATs were developed with the Rasch measurement theory, using full-length CLEFT-Q responses collected during the CLEFT-Q field test (this included 2434 patients across 12 countries). These algorithms were validated in Monte Carlo simulations involving full-length CLEFT-Q responses collected from 536 patients. In these simulations, the CAT algorithms approximated full-length CLEFT-Q scores iteratively, using progressively fewer items from the full-length PROM. Agreement between full-length CLEFT-Q score and CAT score at different assessment lengths was measured using the Pearson correlation coefficient, root-mean-square error (RMSE), and 95% limits of agreement. CAT settings, including the number of items to be included in the final assessments, were determined in a multistakeholder workshop that included patients and health care professionals. A user interface was developed for the platform, and it was prospectively piloted in the United Kingdom and the Netherlands. Interviews were conducted with 6 patients and 4 clinicians to explore end-user experience. ResultsThe length of all 8 CLEFT-Q scales in the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set combined was reduced from 76 to 59 items, and at this length, CAT assessments reproduced full-length CLEFT-Q scores accurately (with correlations between full-length CLEFT-Q score and CAT score exceeding 0.97, and the RMSE ranging from 2 to 5 out of 100). Workshop stakeholders considered this the optimal balance between accuracy and assessment burden. The platform was perceived to improve clinical communication and facilitate shared decision-making. ConclusionsOur platform is likely to facilitate routine CLEFT-Q uptake, and this may have a positive impact on clinical care. Our free source code enables other researchers to rapidly and economically reproduce this work for other PROMs

Recursive partitioning vs computerized adaptive testing to reduce the burden of health assessments in cleft lip and/or palate : comparative simulation study

Background: Computerized adaptive testing (CAT) has been shown to deliver short, accurate, and personalized versions of the CLEFT-Q patient-reported outcome measure for children and young adults born with a cleft lip and/or palate. Decision trees may integrate clinician-reported data (eg, age, gender, cleft type, and planned treatments) to make these assessments even shorter and more accurate. Objective: We aimed to create decision tree models incorporating clinician-reported data into adaptive CLEFT-Q assessments and compare their accuracy to traditional CAT models. Methods: We used relevant clinician-reported data and patient-reported item responses from the CLEFT-Q field test to train and test decision tree models using recursive partitioning. We compared the prediction accuracy of decision trees to CAT assessments of similar length. Participant scores from the full-length questionnaire were used as ground truth. Accuracy was assessed through Pearson’s correlation coefficient of predicted and ground truth scores, mean absolute error, root mean squared error, and a two-tailed Wilcoxon signed-rank test comparing squared error. Results: Decision trees demonstrated poorer accuracy than CAT comparators and generally made data splits based on item responses rather than clinician-reported data. Conclusions: When predicting CLEFT-Q scores, individual item responses are generally more informative than clinician-reported data. Decision trees that make binary splits are at risk of underfitting polytomous patient-reported outcome measure data and demonstrated poorer performance than CATs in this study

Cleft lip and/or palate mortality trends in the USA: a retrospective population-based study

Background Cleft lip and/or palate (CL/P) is one of the most common congenital anomalies worldwide. Although CL/P management may require a series of interventions, mortality resulting from CL/P alone is rare. This study aims to examine recent trends of CL/P mortality rates in the USA.Methods A retrospective population-based study was conducted using official US birth and death certificate data from the Centers for Disease Control and Prevention from 2000 to 2019. Annual mortality rates per 1000 births with CL/P were calculated across sex and racial groups. Multivariable logistic regression models estimated the effects of sex and race on the risk of mortality with CL/P, and linear regression models were used to examine temporal changes in mortality rate across sex and race.Results From 2000 to 2019, 1119 deaths occurred in patients with documented CL/P, for an overall incidence of 20.3 deaths per 1000 births with CL/P (95% CI 18.9 to 22.8). Of these, Patau syndrome was the listed cause of death in 167 cases (14.9%). Black individuals (OR 1.93, 95% CI 1.85 to 2.01), Hispanic (1.54, 1.49 to 1.58) and American Indian individuals (1.28, 1.20 to 1.35) were at a greater risk of CL/P mortality compared with white individuals. Additionally, females were also at a greater risk (1.35, 1.21 to 1.49). A significant upward trend in CL/P mortality was observed in Hispanic (r2=0.70, p<0.01) and American Indian individuals (r2=0.81, p<0.01) from 2000 to 2019.Conclusions Cleft birth and mortality surveillance is essential in healthcare and prevention planning. Future studies are required to understand the differences in CL/P mortality rates across various sociodemographic groups

The First Alveolar Bone Graft Simulator

Summary:. Alveolar bone graft (ABG) surgery in cleft patients is technically challenging. The procedure requires design, dissection and release of soft tissue flaps to create a seal around the bone graft. In addition, visualization during the procedure is challenging within the confines of the cleft. These features make ABG surgery difficult to learn and teach, and it is, therefore, a suitable procedure for the use of a simulator. A high-fidelity cleft ABG simulator was developed using three-dimensional printing, polymer, and adhesive techniques. Simulated ABG surgery was performed by two expert cleft surgeons for a total of five simulation sessions to test the simulator’s features and the ability to perform the critical steps of an ABG. ABG surgery was successfully performed on the simulator. The simulations involved interacting with realistic dissection planes as well as multi-layered synthetic soft (periosteum, mucosa, gingiva, adipose tissue) and hard (teeth, bone) tissue. The simulator allowed performance of cleft marginal incisions, dissection, and elevation of a muco-gingival-periosteal flap, creation of nasal upturned and palatal downturned flaps, nasal and palatal side closure, insertion of simulated bone graft material, and advancement of the muco-gingival-periosteal flap for closure of the anterior wall of the cleft. The ABG simulator allowed performance of the critical steps of ABG surgery. This is the first ABG simulator developed, which incorporates the features necessary to practice the procedure from start to finish

A High Fidelity Cleft Lip Simulator

Background:. Cleft lip surgery is technically difficult requiring precise planning and understanding of 3-dimensional structures to obtain an optimal outcome. A physical cleft lip simulator was developed that allows trainees to gain experience in cleft lip repair and primary rhinoplasty before operating on real patients. Methods:. A cleft lip simulator that comprises multilayered soft tissues, bone, and realistic dissection planes was developed using 3D printing, adhesive and polymer techniques. Four experienced cleft surgeons performed a total of 7 simulated repairs on the simulator. Feedback on the realism and value of the simulator was obtained from the surgeons. Results:. Six of the repairs were a Fisher anatomic subunit approximation technique, and 1 was a rotation advancement repair. All repairs were completed with successful performance of markings, incisions, dissections, and multilayered closure. All surgeons agreed that the simulator is realistic and that the simulator is a valuable tool for training in cleft lip surgery. Conclusions:. A cleft lip simulator that allows performance of a cleft lip repair and primary rhinoplasty from start to finish was developed and pilot tested. The simulator provides a training platform to gain experience in cleft lip repair before operating on real patients

sj-docx-1-cpc-10.1177_10556656241230882 - Supplemental material for Bilateral Cleft lip Simulation

Supplemental material, sj-docx-1-cpc-10.1177_10556656241230882 for Bilateral Cleft lip Simulation by Jonathan Zaga-Galante, Raymond Tse, Richard A Hopper, Anne Arnold, David M Fisher, Karen W Wong-Riff and Dale J Podolsky in The Cleft Palate Craniofacial Journal</p

sj-docx-6-cpc-10.1177_10556656241230882 - Supplemental material for Bilateral Cleft lip Simulation

Supplemental material, sj-docx-6-cpc-10.1177_10556656241230882 for Bilateral Cleft lip Simulation by Jonathan Zaga-Galante, Raymond Tse, Richard A Hopper, Anne Arnold, David M Fisher, Karen W Wong-Riff and Dale J Podolsky in The Cleft Palate Craniofacial Journal</p

sj-docx-2-cpc-10.1177_10556656241230882 - Supplemental material for Bilateral Cleft lip Simulation

Supplemental material, sj-docx-2-cpc-10.1177_10556656241230882 for Bilateral Cleft lip Simulation by Jonathan Zaga-Galante, Raymond Tse, Richard A Hopper, Anne Arnold, David M Fisher, Karen W Wong-Riff and Dale J Podolsky in The Cleft Palate Craniofacial Journal</p