Cross-cultural adaptation of the Thai Xerostomia Inventory and Summated Xerostomia Inventory.

OBJECTIVE: To cross-culturally adapt and validate the Thai version of the Xerostomia Inventory (XI) and Summated Xerostomia Inventory (SXI) for subjective evaluation of oral dryness in Thai middle-aged and older adults. METHODS: The original English versions of the XI and SXI were cross-culturally translated into Thai. Content validity was examined by the expert panel and 30 pilot subjects. The XI/SXI-Thai were then administered to 200 Thai adults (aged 50 years or older) for further assessment of psychometric properties. The standard question, the Bother Index, and the Xerostomia Questionnaire were used as comparator instruments for the evaluation of criterion and construct validity, respectively. Internal consistency reliability was assessed using Cronbach's alpha. RESULTS: The final XI/SXI-Thai were found to have good content validity. Both scales were able to distinguish between xerostomia and non-xerostomia groups. We observed moderate to strong correlation between the XI/SXI-Thai and other comparator instruments, reflecting good criterion and construct validity. Cronbach's alpha values were 0.875 and 0.847 for the XI-Thai and SXI-Thai, respectively. CONCLUSIONS: The XI-Thai and SXI-Thai were found to be valid, reliable, and easily administrable instruments for xerostomia assessment in Thai middle-aged and older individuals in both clinical and research settings.

World Workshop on Oral Medicine VIII: Development of a core outcome set for dry mouth: a systematic review of outcome domains for salivary hypofunction.

OBJECTIVE: To identify all outcome measures used to assess salivary gland hypofunction (i.e., objective measures used to determine actual changes in saliva quantity or to assess response to treatment of salivary gland hypofunction) and to group these into domains. STUDY DESIGN: A systematic review including clinical trials and prospective or retrospective observational studies involving human participants with dry mouth, with any type of intervention where the objective assessment of salivary gland hypofunction was described. RESULTS: Five hundred fifty-three studies involving 31,507 participants were identified. Most assessed salivary gland hypofunction and xerostomia (68.7%), whereas 31.3% assessed salivary gland hypofunction alone. Most studies investigated the "amount of saliva," and the highest number of outcome measures were within the domain of "clinical/objective signs of salivary gland hypofunction." CONCLUSIONS: Seven domains encompassing 30 outcome measures were identified, confirming the diversity in outcomes and outcome measures used in research regarding salivary gland hypofunction. Identified items will be used in conjunction with those identified regarding xerostomia to create a core outcome set for dry mouth quantification for use in future clinical trials, with the overall goal of improving the standardization of reporting, leading to the establishment of more robust evidence for the management of dry mouth and improving patient care.

World Workshop on Oral Medicine VIII: Development of a core outcome set for dry mouth: a systematic review of outcome domains for xerostomia.

OBJECTIVE: The purpose of this study was to identify all outcome domains used in clinical studies of xerostomia, that is, subjective sensation of dry mouth. This study is part of the extended project "World Workshop on Oral Medicine Outcomes Initiative for the Direction of Research" to develop a core outcome set for dry mouth. STUDY DESIGN: A systematic review was performed on MEDLINE, EMBASE, CINAHL, and Cochrane Central Register of Controlled Trials databases. All clinical and observational studies that assessed xerostomia in human participants from 2001 to 2021 were included. Information on outcome domains was extracted and mapped to the Core Outcome Measures in Effectiveness Trials taxonomy. Corresponding outcome measures were summarized. RESULTS: From a total of 34,922 records retrieved, 688 articles involving 122,151 persons with xerostomia were included. There were 16 unique outcome domains and 166 outcome measures extracted. None of these domains or measures were consistently used across all the studies. The severity of xerostomia and physical functioning were the 2 most frequently assessed domains. CONCLUSION: There is considerable heterogeneity in outcome domains and measures reported in clinical studies of xerostomia. This highlights the need for harmonization of dry mouth assessment to enhance comparability across studies and facilitate the synthesis of robust evidence for managing patients with xerostomia.

World Workshop on Oral Medicine VIII: Development of a core outcome set for dry mouth: a consensus study.

OBJECTIVE: This study aimed to develop a consensus-based core outcome set (COS) to be used in clinical trials assessing dry mouth interventions. STUDY DESIGN: Through 2 systematic literature reviews and interviews with dry mouth patients, we identified relevant outcome domains for dry mouth assessment. A Delphi survey was presented to health care providers attending the American Academy of Oral Medicine annual meeting in Memphis, Tennessee, USA, on May 2022 (n = 104) and 10 dry mouth patients at Cork University Dental School and Hospital, Republic of Ireland. The outcome domains for which no consensus was reached were subsequently discussed in a second consensus process led by a virtual Special Interest Group of 11 oral medicine experts from the World Workshop on Oral Medicine VIII dry mouth working group. RESULTS: After the 2-step consensus process, a consensus was reached for 12 dry mouth outcome domains (i.e., salivary gland flow, signs of hyposalivation, mucosal moisture/wetness, the severity of xerostomia, duration of xerostomia, the overall impact of xerostomia, impact on physical functioning, impact of hyposalivation on general health, impact on social activities, quality of life, the economic impact of dry mouth, patient satisfaction) to be included in the final COS. CONCLUSIONS: We propose a consensus-based COS to assess dry mouth interventions in clinical trials. This COS includes the minimum but mandatory set of domains that all clinical trials evaluating dry mouth treatments should assess.

A Mutation in CACNA1S Is Associated with Multiple Supernumerary Cusps and Root Maldevelopment.

BACKGROUND: Enamel knots and Hertwig epithelial root sheath (HERS) regulate the growth and folding of the dental epithelium, which subsequently determines the final form of tooth crown and roots. We would like to investigate the genetic etiology of seven patients affected with unique clinical manifestations, including multiple supernumerary cusps, single prominent premolars, and single-rooted molars. METHODS: Oral and radiographic examination and whole-exome or Sanger sequencing were performed in seven patients. Immunohistochemical study during early tooth development in mice was performed. RESULTS: A heterozygous variant (c. 865A>G; p.Ile289Val) in CACNA1S was identified in all the patients, but not in an unaffected family member and control. Immunohistochemical study showed high expression of Cacna1s in the secondary enamel knot. CONCLUSIONS: This CACNA1S variant seemed to cause impaired dental epithelial folding; too much folding in the molars and less folding in the premolars; and delayed folding (invagination) of HERS, which resulted in single-rooted molars or taurodontism. Our observation suggests that the mutation in CACNA1S might disrupt calcium influx, resulting in impaired dental epithelium folding, and subsequent abnormal crown and root morphology.

Validity and responsiveness of pain rating scales in patients with chronic oral mucosal diseases.

OBJECTIVES: To validate the Visual Analog Scale (VAS) and Numerical Rating Scale (NRS) for measuring pain intensity in chronic oral mucosal diseases. METHODS: Secondary analyses of data including the VAS, NRS, demographic, clinical and quality-of-life outcomes at baseline and 4-month follow-up were retrieved from a clinical study of chronic oral mucosal diseases. Construct and criterion validity and responsiveness of the VAS and NRS were assessed through testing hypotheses based upon strength of Spearman's correlation coefficients. RESULTS: Data of 500 and 290 patients with chronic oral mucosal diseases were used for the assessment of validity and responsiveness, respectively. Moderate-to-high correlations between both pain scores and scores of clinical and quality-of-life outcomes were observed, supporting construct validity of the VAS and NRS. Their criterion validity was confirmed by significantly strong association between scores of both scales. Responsiveness of both scales was adequate based on moderate association between their change scores and global rating of change scale. CONCLUSION: The present results provide evidence supporting validity and responsiveness of the VAS and NRS for pain intensity assessment in patients with chronic oral mucosal diseases. Future research examining other pain intensity domains and standardizing composite scores for pain intensity in this population is required.

Evaluating the measurement properties of patient-reported outcome measures in radiotherapy-induced xerostomia.

OBJECTIVE: Radiotherapy-induced xerostomia (RIX) is one of the most common adverse effects of radiotherapy to the head and neck, and a major determinant of survivors' quality of life. A number of patient-reported outcome measures (PROMs) have been used in clinical trials of therapeutic interventions for RIX; however, little is known regarding their measurement properties and methodological quality. METHODS: We conducted a systematic literature search in Embase, MEDLINE and PsycINFO for articles published up to May 2019 and evaluating at least one measurement property of PROMs relevant to RIX. The COSMIN guidelines were used to assess relevant measurement properties and methodological quality. RESULTS: Nine validations studies were identified reporting on four PROMs relevant to RIX. The Xerostomia Questionnaire (XQ) showed overall high-quality evidence for structural validity and internal consistency, but low-quality evidence supporting reliability. The methodological quality of the Groningen Radiotherapy-Induced Xerostomia scale (GRIX), Xerostomia Inventory (XI) and the Xerostomia Quality of Life Scale (XeQoLS) was relatively low for all measurement properties. CONCLUSIONS: The XQ was found to have the highest potential to capture changes in RIX according to COSMIN guidelines. Additional validation studies are required to further understand the methodological quality of the XI, GRIX and XeQoLS.

The patient acceptable symptom state in oral lichen planus: identification of cut-off threshold scores in measures of pain and quality of life.

OBJECTIVES: To establish thresholds of pain and quality of life scores corresponding to patient acceptable symptom state (PASS) in patients with oral lichen planus (OLP) and to assess demographic and clinical factors associated with achieving the PASS. METHODS: Prospective data from baseline and 4-month follow-up including Visual Analogue Scale (VAS), Numerical Rating Scale (NRS), 14-item Oral Health Impact Profile (OHIP-14) and 15-item and 26-item Chronic Oral Mucosal Disease Questionnaire (COMDQ-15; COMDQ-26) were collected from 281 patients with OLP. An anchoring approach based upon the patient's opinion on acceptability of OLP status was applied. Associated factors for achieving the PASS were analysed using multivariate logistic regression. RESULTS: About two-thirds (68.7%) of participants rated their OLP status as acceptable. Cut-off thresholds for PASS were as follows: ≤ 28 mm for VAS, ≤ 3 for NRS, ≤ 18 for total OHIP-14, ≤ 26 for total COMDQ-15 and ≤ 48 for total COMDQ-26. Based upon results of multivariate logistic analysis, factors associated with being in PASS were lower pain intensity, lower depressive symptoms and lower disease activity of OLP. CONCLUSION: The present study established PASS cut-off thresholds as a tool facilitating interpretation of pain and quality of life outcomes relevant to individuals with OLP. CLINICAL RELEVANCE: Identified PASS estimates could be utilised as clinically important endpoints in clinical practice of OLP as well as eligibility criteria for recruiting participants in clinical trials assessing effectiveness of symptomatic intervention of OLP.

Health-related quality of life and its associated predictors in patients with oral lichen planus: a cross-sectional study.

OBJECTIVES: To investigate levels of quality of life (QoL) and determine associated predictors in patients with oral lichen planus (OLP). MATERIALS AND METHODS: A total of 300 patients with OLP at one tertiary Oral Medicine clinic in the UK were recruited in a cross-sectional study from January 2018 to July 2019. The 15-item Chronic Oral Mucosal Disease Questionnaire (COMDQ-15) and 14-item Oral Health Impact Profile (OHIP-14) were used to assess the level of QoL related to OLP. A number of potential determinants were considered, including patient demographics, treatment, the severity of oral symptoms, the clinical activity of the disease, and the patient psychological status, which were measured using the pain-Numerical Rating Scale, the Oral Disease Severity Score, the Hospital Anxiety and Depression Scale, and the 10-item Perceived Stress Scale. Multivariate linear regression was employed to identify independent determinants associated with overall and aspects of QoL. RESULTS: On multivariate analyses, after adjusting for confounding variables, the QoL levels in patients with OLP were significantly associated with levels of oral pain, anxiety, stress and use of topical corticosteroids. The COMDQ-15 instrument performed better than OHIP-14 at capturing the association between QoL and pain and disease activity in patients with OLP. CONCLUSION: Clinicians should expect reduced QoL in OLP patients with high pain levels, high anxiety levels, high perceived stress and use of topical corticosteroids. The COMDQ-15 is best suited to measure QoL in this population.

Meaningful improvement thresholds in measures of pain and quality of life in oral lichen planus.

OBJECTIVES: To evaluate the responsiveness of measures of pain and oral health-related quality of life (OH-QoL) in patients with oral lichen planus (OLP) and to determine thresholds for minimal important change (MIC) and minimal important difference (MID) for use in this patient population. METHODS: Data from baseline and 4-month follow-up including Visual Analog Scale (VAS), Numerical Rating Scale (NRS), 14-item Oral Health Impact Profile (OHIP-14), 15-item and 26-item Chronic Oral Mucosal Disease Questionnaire (COMDQ-15; COMDQ-26) were collected from 157 patients with OLP. Responsiveness was assessed by testing hypotheses and calculating the area under the curve. MIC and MID were established based on triangulation of distribution-based and anchor-based estimates. RESULTS: The results supported adequate responsiveness of VAS, NRS, COMDQ-15 and COMDQ-26 for use in OLP, while the OHIP-14 demonstrated relatively low sensitivity to detect improvement in the OLP status. Recommended meaningful improvement thresholds were as follows: VAS (MIC 16 mm; MID 18 mm), NRS (MIC/MID 2 points), OHIP-14 (MIC/MID 5 points), COMDQ-15 (MIC 5 points; MID 6 points) and COMDQ-26 (MIC/MID 9 points). CONCLUSION: This study provides some evidence of responsiveness as well as establishing meaningful improvement thresholds in scores of pain and OH-QoL measures in OLP.

Validation of the HADS and PSS-10 and a cross-sectional study of psychological status in patients with recurrent aphthous stomatitis.

OBJECTIVES: To validate the Hospital Anxiety and Depression Scale (HADS) and the 10-item Perceived Stress Scale (PSS-10) for use in clinical studies of recurrent aphthous stomatitis (RAS) and to provide cross-sectional assessment of anxiety, depressive, and distress symptoms and perceived stress in patients with RAS. METHODS: The validity and reliability of the HADS and PSS-10 were evaluated in 120 individuals with RAS through confirmatory factor analysis and calculation of Cronbach's alpha and omega coefficients. The prevalence of comorbid anxiety, depression, distress, and moderate-to-high perceived stress, and their association with demographics and clinical factors were assessed through cutoff scores of the HADS and PSS-10 and bivariate analyses, respectively. RESULTS: A bi-factor model, with all items loading onto general factor with two group factors, provides the best fit to the HADS and PSS-10 data of this RAS cohort. While omega values suggested adequate reliability of total score of both scales, relatively low ranges of coefficient omega hierarchical limit utility of their subscale scores. The prevalence of anxiety, depression, distress, and moderate-to-high perceived stress was 42.5%, 18.33%, 28.33%, and 71.67%, respectively. Ethnicity, alcohol consumption, disease comorbidities, clinical type of RAS, ulcer size, pain, and RAS disease activity were found to be associated with negative psychological symptoms. CONCLUSION: The HADS and PSS-10 are valid and reliable as general scales of psychological distress and stress in patients with RAS. Significant mental burden among RAS patients makes the use of these validated instruments a sensible and prudent practice for psychological assessment of this patient group.

Development and validation of a short version of Chronic Oral Mucosal Disease Questionnaire (COMDQ-15).

BACKGROUND: The adoption of the Chronic Oral Mucosal Disease Questionnaire (COMDQ) into clinical practice has been low, despite its rigorous development process. A potential limitation of the COMDQ is the high response burden to patients. Therefore, the aim of the present study was to develop and validate a short version of the 26-item COMDQ. METHODS: The COMDQ data of 520 patients with chronic oral mucosal diseases were randomly divided into two subsamples. Descriptive item analysis and exploratory factor analysis (EFA) were performed using data from the first subsample for item reduction and development of the shortened COMDQ. The resulting short version was then validated using confirmatory factor analysis (CFA) on the other subsample. Internal consistency reliability of the short-form COMDQ was assessed using Cronbach's alpha. Criterion validity of this new scale was examined against its original version. RESULTS: Based upon item analysis, 11 items were dropped. EFA results on the remaining 15 items extracted four factors consistent with the original COMDQ, and CFA results displayed acceptable goodness-of-fit indices of this factor structure on different sample. The COMDQ-15 was then created. Cronbach's alpha of four subscale scores ranged from 0.7 to 0.91, indicating good internal consistency reliability of the COMDQ-15. Correlations between total and subscale scores of the COMDQ-15 and its parent scale were high, supporting good criterion validity of this shortened scale. CONCLUSION: The COMDQ-15 is a brief, valid and reliable instrument that can give an overview of the patient's quality of life related to their chronic oral mucosal conditions.

Validation of the HADS and PSS-10 and psychological status in patients with oral lichen planus.

OBJECTIVES: To validate the Hospital Anxiety and Depression Scale (HADS) and the 10-item Perceived Stress Scale (PSS-10) for use in patients with oral lichen planus (OLP) and to provide cross-sectional analysis of anxiety, depressive and distress symptoms in patients with OLP. METHODS: Validity and reliability of both instruments were assessed in 260 participants with OLP in one tertiary oral medicine centre through confirmatory factor analysis and calculation of reliability coefficients. Prevalence, clinical and demographic predictors of the presence of psychological symptoms in OLP were calculated and identified using multivariated logistic regression. RESULTS: Factor analysis results demonstrated that a bifactor model described the underlying structure of both scales better than other models. Values of omega indicated adequate reliability of total HADS and PSS-10 score while low coefficient omega hierarchical values limit clinical applicability of their subscale scores. The prevalence of anxiety, depressive and distress symptoms in OLP was 39.23%, 20.77% and 27.69%, respectively. Pain intensity, disease comorbidities, age, smoking and alcohol consumption were found to be independent predictors of the presence of psychological symptoms in OLP. CONCLUSION: The HADS and PSS-10 are appropriate to use as general measures of psychological distress and perceived stress in patients with OLP.

Measurement properties of patient-reported outcome measures in radiotherapy-induced trismus.

Trismus or lockjaw is the reduction in mouth opening. Radiotherapy-induced trismus occurs when the masticatory muscles are within range of radiation treating head and neck cancer, resulting in muscular fibrosis and trismus. We aim to assess the measurement properties of available validated patient-reported outcome measures relevant to radiotherapy-induced trismus in head and neck cancer patients. Systematic electronic searches were performed in MEDLINE and EMBASE databases to identify studies validating radiotherapy-induced trismus with patient-reported outcome measures up to January 2018. Two reviewers independently rated the methodological quality of identified validation studies, using the consensus-based standards for the selection of health status measurement instrument (COSMIN) checklist. The level of evidence for measurement properties was then summarised based upon pre-defined criteria. Only one of the 280 retrieved studies were deemed fit for inclusion. The Gothenburg Trismus Questionnaire was assessed and found to have a good methodological quality of measurement properties except for reliability, which was poor. There was a moderate level of evidence supporting internal consistency, content validity, structural validity and hypothesis testing. The evidence for test-retest reliability remains unknown in the population of interest. The Gothenburg Trismus Questionnaire is the only patient-reported outcome measure in the field of showing some good measurement properties, and it may therefore represent a useful instrument for the assessment of radiotherapy-induced trismus in head and neck cancer patients. However, further validation studies are needed especially with respect to the reliability, measurement error and the responsiveness of this instrument.

Patient-reported outcome measures in oral lichen planus: A comprehensive review of the literature with focus on psychometric properties and interpretability.

OBJECTIVE: To review the range of patient-reported outcome measures (PROMs) used in clinical studies of patients with oral lichen planus (OLP) and to assess their psychometric properties and interpretability. METHODS: Literature searches were performed on MEDLINE, EMBASE and Web of Science databases (1990-September 2016) to retrieve relevant studies related to the development, psychometric testing and/or use of PROMs assessing oral symptoms, psychosocial status and quality of life in individuals with OLP. The identified PROMs were then categorised by concept measured and assessed for instrument characteristics and evidence for psychometric properties and interpretability. RESULTS: We identified a total of 41 PROMs used in clinical studies for the assessment of patient-reported outcomes in patients with OLP. There were three PROMs of oral symptoms, 30 PROMs of psychosocial status and eight PROMs of quality of life. Six instruments (Visual Analog Scale, Numerical Rating Scale, Change in Symptom Scale, Oral Health Impact Profile-14, Oral Health-related Quality of Life-UK and Chronic Oral Mucosal Disease Questionnaire) demonstrated some evidence of psychometric properties but no evidence for interpretability of their results in the OLP population. CONCLUSION: The range of PROMs used in clinical studies of patients with OLP is wide and include instruments for oral symptoms, psychosocial status and quality of life. The vast majority of these instruments have no evidence of psychometric properties and interpretability for patients with OLP. Further qualitative and validation studies are required to investigate whether these instruments are appropriate for use in this patient population.

Web-based information on the treatment of oral leukoplakia - quality and readability.

OBJECTIVE: To categorise the content and assess the quality and readability of the online information regarding the treatment for oral leukoplakia. METHODS: An online search using the term 'leukoplakia treatment' was carried out on 8th June 2015 using the Google search engine. The content, quality and readability of the first 100 sites were explored. The quality of the web information was assessed using the following tools, the DISCERN instrument and the Journal of the American Medical Association (JAMA) benchmarks for website analysis and the HON seal. Readability was assessed via the Flesch Reading Ease Score. RESULTS: The search strategy generated 357 000 sites on the Google search engine. Due to duplicate links, non-operating links and irrelevant links, a total of 47 of the first 100 websites were included in this study. The mean overall rating achieved by included websites using the DISCERN instrument was 2.3. With regard to the JAMA benchmarks, the vast majority of examined websites (95.7%) completely fulfilled the disclosure benchmark and less than 50% of included websites met the three remaining criteria. A mean total readability score of 47.5 was recorded with almost 90% of websites having a readability level ranging from fairly difficult to very difficult. CONCLUSION: Based on this study, the online health information regarding oral leukoplakia has challenging readability with content of questionable accuracy. As patients often search for health information online, it would be prudent for clinicians to highlight the caution with which online information should be interpreted.