The Laryngoscope C 2017 The American Laryngological, V Rhinological and Otological Society, Inc. TRIOLOGICAL SOCIETY CANDIDATE THESIS Development of Cognitive Screening Test for the Severely Hearing Impaired: Hearing-Impaired MoCA Vincent Y. W. Lin, MD, FRCSC; Janet Chung, MD, FRCSC; Brandy L. Callahan, PhD, Psych; Leah Smith, MA, CCRA; Nils Gritters, BSc (Hons); Joseph M. Chen, MD, FRCSC; Sandra E. Black, OC, O.Ont. MD, FRSC, FAAN, FANA; Mario Masellis, MSc, MD, PhD, FRCPC Objectives: To develop a version of the Montreal Cognitive Assessment (MoCA) to be administered to the severely hearing impaired (HI-MoCA), and to assess its performance in two groups of cognitively intact adults over the age of 60. Study Type: Test development followed by prospective subject recruitment. Methods: The MoCA was converted into a timed PowerPoint (Microsoft Corp., Redmond, WA) presentation, and verbal instructions were converted into visual instructions. Two groups of subjects over the age of 60 were recruited. All subjects passed screening questionnaires to eliminate those with undiagnosed mild cognitive impairment. The first group had normal hearing (group 1). The second group was severely hearing impaired (group 2). Group 1 received either the MoCA or HI-MoCA test (T1). Six months later (T2), subjects were administered the test (MoCA or HI-MoCA) they had not received previously to determine equivalency. Group 2 received the HI-MoCA at T1 and again at T2 to determine test–retest reliability. Results: One hundred and three subjects were recruited into group 1, with a score of 26.66 (HI-MoCA) versus 27.14 (MoCA). This was significant (P < 0.05), but scoring uses whole numerals and the 0.48 difference was found not clinically significant using post hoc sensitivity analyses. Forty-nine subjects were recruited into group 2. They scored 26.18 and 26.49 (HI-MoCA at T1 and T2). No significance was noted (P > 0.05), with a test–retest coefficient of 0.66. Conclusion: The HI-MoCA is easy to administer and reliable for screening cognitive impairment in the severely hearing impaired. No conversion factor is required in our prospectively tested cohort of cognitively intact subjects. Key Words: hearing loss, dementia, screening, elderly. Level of Evidence: 1b. Laryngoscope, 127:S4–S11, 2017 INTRODUCTION The association between hearing loss and dementia was first proposed in 1989 in a case-control study published by Uhlmann et al. in the Journal of the American Medical Association.1 In this article, 100 patients with Alzheimer’s-type dementia were matched to 100 control subjects without dementia with respect to age, sex, and education. They found that hearing loss was significantly and independently correlated with the severity of cognitive dysfunction. This important relationship was From the Department of Otolaryngology–Head & Neck Surgery (V.Y.W.L., JANET C., L.S., N.G., JOSEPH C.); the Division of Neurology (B.L.C., S.E.B., M.M.); the Sunnybrook Research Institute (V.Y.W.L., B.L.C., L.S., N.G., JOSEPH C., S.E.B., M.M.), Sunnybrook Health Sciences Centre; and the Department of Otolaryngology–Head & Neck Surgery, Faculty of Medicine, University of Toronto (V.Y.W.L., JANET C., JOSEPH C.), Toronto, Ontario, Canada. Editor’s Note: This Manuscript was accepted for publication February 21, 2017. This study was partially funded by MED-EL, GmbH via a research grant to the primary author (V.Y.W.L.). The authors have no other funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Vincent Y.W. Lin, MD, FRCSC, Associate Professor, Associate Scientist, Department of Otolaryngology–Head & Neck Surgery, Sunnybrook Research Institute, Sunnybrook Health Sciences Centre, Institute of Medical Sciences, Faculty of Medicine, University of Toronto, M1-102, 2075 Bayview Avenue, Toronto, Ontario, M4N 3M5. E-mail: [email protected] sparsely studied until 2011, when Lin et al. released a series of groundbreaking publications. Their work established that age-related hearing loss is independently associated with worse cognitive function2 and incidental dementia.3 They also established that the severity of hearing loss correlates with a higher risk of developing dementia; those with mild, moderate, and severe hearing loss, respectively, had a 1.9-, 3.0-, and 4.9-fold increased risk of developing dementia. Although an etiological link has yet to be determined, an important question remains: how do we best identify hearing-impaired patients who are at risk of developing, or who already have, early dementia? A screening measure, appropriate for the hearing-impaired population, would allow for earlier environmental and pharmacological intervention in individuals with early cognitive impairment/dementia. It also would facilitate identification of such patients for studies attempting to unravel this etiological link. Prevalence and Cost of Dementia DOI: 10.1002/lary.26590 In Canada, the prevalence of dementia is rising due to the rapidly growing aging segment of the population. Wong et al. used information from the 2010/2011 Canadian Community Health Survey, the 2011/2012 Survey of Neurological Conditions in Institutions in Canada, and the 2011 Survey on Living with Neurological Conditions in Canada and calculated that for individuals over the age Laryngoscope 127: 2017 Lin et al.: Development of Hearing-Impaired MoCA S4 of 45, 0.8% of those living in private households and 45% of patients in long-term residential care facilities had a diagnosis of dementia.4 There was an estimated 53,100 patients living in long-term residential care with dementia, and the prevalence rose with increasing age. The impending socioeconomic costs of dementia have been long recognized. In an article published in 1994, Ostbye et al. calculated the net cost of dementia in Canada to be 3.9 billion dollars.5 Those costs included the cost of care in long-term institutions and medical/social assistance. More recently in 2011, the Alzheimer’s Society of Canada calculated the cost in 2011 to be $33 billion a year. This cost was estimated to rise to $293 billion a year by 2040. Caregiver Burden As the severity of dementia in an individual advances, there is an increasing reliance on caregivers for maintaining their activities of daily living. This usually falls upon immediate family members, but as dementia worsens and other complications (e.g., immobility and behavioral disturbances) emerge, patients are often transferred to long-term care facilities. In Canada, family caregivers in up to 90% of these situations provide in-home care for people with dementia.6 In the United States, there are over 15 million unpaid caregivers who collectively provide 17.9 billion hours of assistance to the estimated 5.3 million patients with Alzheimer’s disease (AD).7 There undoubtedly is a large amount of burden on the caregivers of dementia patients both personally and economically.8,9 It appears that caregiver burden is not only a first-world issue, Pattanayak et al. also describe a similar increasing concern regarding caregiver burden in India.10 Diagnosis of Dementia Although the prevalence of dementia is rising, its diagnosis still can remain challenging, especially in individuals with primary sensory deficits. A diagnosis of dementia, also called major neurocognitive disorder, requires subjective and objective evidence of cognitive decline sufficiently marked to impact activities of daily living.11 In patients with a high likelihood of dementia due to AD, this decline must affect at least two of the following domains: 1) memory, 2) communication/ language, 3) attentiveness/focus on a program or subject, 4) reasoning/judgment, and 5) visual perception.12 A recent systematic review performed by Ngo et al. reviewed dementia practice guidelines from 12 working groups from a variety of countries, including Canada, the United States, the United Kingdom, Korea, Australia, Singapore, Malaysia, and European countries.13 General consensus was found in four main areas: 1) cognitive testing, 2) comorbid conditions, 3) clinical diagnosis, and 4) imaging/specialized tests. Some highlights include testing cognition using only standardized screening tools, such as the Montreal Cognitive Assessment (MoCA); running a standard battery of bloodwork, including complete blood count, thyroid-stimulating hormone, electrolytes, calcium, fasting blood glucose, vitamin B12, and folate; making diagnoses of dementia using internationally agreed upon consensus criteria; and using at least one imaging Laryngoscope 127: 2017 modality. Although consensus was seen in a number of guideline recommendations, diverging recommendations were still evident in their review. Clearly the diagnosis of dementia and its various subtypes requires the skill of experienced clinicians relying on a wide breadth of standardized tests to aid them. Diagnosis of Mild Cognitive Impairment Substantial effort has been made in recent years to move toward earlier diagnosis and disease management. Mild cognitive impairment (MCI), also called mild neurocognitive disorder by Diagnostic Statistical Manual of Mental Disorders-V criteria,11 describes cognitive decline that is beyond expected age-related change and is assumed to reflect incipient dementia, although not severe enough to interfere with independence in functional abilities.14 Core clinical criteria include: 1) concern regarding a change in cognition, 2) impairment in one or more cognitive domains, 3) preservation of independence in functional abilities, and 4) not demented with no evidence of significant impairment in social or occupational functioning.14 MCI is of particular interest to clinicians not only because these patients can still function at a fairly high level but because it is felt that interventions may be most effective in these early-stage patients.15 According to the Alzheimer’s Society of Canada: Family, friends and the person with MCI may notice these changes and they can be objectively measured in cognitive tests, but the changes in cognitive abilities are usually not serious enough to interfere with daily life and independence. A person with MCI has an increased risk of developing AD or another dementia, but some people with MCI remain stable and others show improved cognitive abilities over time. There is no single cause or outcome for people diagnosed with MCI. A wide range of cognitive abilities can be impaired, but for the majority of people who have MCI memory is the cognitive ability most affected. Superiority of MoCA The gold standard to identify cognitive changes that may be indicative of dementia remains formal neuropsychological assessment involving the standardized administration of a broad battery of measures by a trained professional.16 To identify individuals who may benefit from such in-depth neuropsychological testing, panels of experts worldwide have issued formal recommendations for general practitioners to screen for dementia in highrisk groups.17 Historically, the Mini-Mental State Examination (MMSE) has been widely used for cognitive screening. It is brief and easy to administer and score. Generally, a score of 26 or greater out of 30 is considered normal, taking into account education level. However, multiple studies have demonstrated the MMSE’s low sensitivity in the screening of cognitive impairment, especially in those with mild cognitive impairment MCI.17–19 Indeed, the vast majority of patients with a neuropsychologically confirmed clinical diagnosis of MCI score Lin et al.: Development of Hearing-Impaired MoCA S5 above the normal cutoff of 26 on the MMSE.20 The MoCA was developed as a more sensitive screening tool for patients with MCI and also is useful for more severe stages of dementia. The MoCA also is easy to administer and score, with normal subjects scoring 26 or greater out of a total score of 30 (www.mocatest.org).20 It consists of 11 distinct test components measuring executive functions, language, attention, abstraction, verbal fluency, calculation, concentration, visuospatial skills, orientation, and delayed recall. Validation studies involved normal controls, patients with confirmed MCI, and those with confirmed AD.19,20 Patients in all groups were tested with both the MMSE and MoCA, and the results indicated that the MoCA’s sensitivity in detecting MCI was 90%, far superior to the MMSE’s sensitivity (18%). The authors attributed the MoCA’s significant advantage to its testing of executive functions, higher-level language abilities, and complex visuospatial processing. It also has been used effectively to identify mild cognitive impairment and dementia in patients with Parkinson’s disease.21 Need for HI-MoCA It has been demonstrated that healthy community living adults (mean age 71) with hearing impairment perform more poorly on the MoCA, even if the hearing impairment is not severe enough to prohibit standard verbal administration of the test.22 Severe hearing impairment clearly prevents most forms of standardized neuropsychological test administration from being properly and accurately administered. Thus, test performances are negatively impacted.23 For patients with severe hearing impairment, the administration of the MoCA as a sensitive screening test for MCI and dementia is currently impossible given that the test requires verbal instructions. In our adult cochlear implantation program, there has been a substantial increase in the number of older patients (> 55 years old) who are being considered for cochlear implantation, primarily due to the expansion of implantation criteria and the well-documented aging of our population. Informally during consultation by either the cochlear implant audiologist or surgeon, the patient’s family may raise concerns regarding the possibility of cognitive impairment, or it may be noted by the clinician during the encounter. In these circumstances, no validated and recognized screening test for cognitive impairment exists that could be used to assess severely hearing-impaired individuals. Furthermore, it is critical to identify if a patient suffers from MCI or dementia because outcomes after surgery often are worse for this subgroup of elderly individuals from the perspective of developing delirium and postoperative complications.24 Therefore, out of this clinical need, we sought to adapt the MoCA for hearing-impaired patients (HI-MoCA) and validate this modified test first in our group of cognitively intact subjects. Study Objectives Our study objectives were to develop a version of the MoCA that could be administered to the severe Laryngoscope 127: 2017 S6 hearing impaired, namely the hearing-impaired MoCA (HI-MoCA), and to validate it in two groups of cognitively intact adults over the age of 60. Study Aims 1. Develop a hearing-impaired version of the MoCA: the HI-MoCA. a. Convert the verbal instructions into visual instructions displayed on an interactive Microsoft Powerpoint (Microsoft Corp., Redmond, WA) presentation that is as internally consistent as possible with the original MoCA. b. Have the HI-MoCA administered without any verbal cues so the tester is present simply to ensure that the proper visual directions are being followed. 2. Validate the HI-MoCA in two groups of cognitively intact individuals. Cognition would be assessed with a validated battery of cognitive screening tests. a. Group 1: Normal hearing, cognitively intact b. Group 2: Severe hearing loss, cognitively intact Study Hypothesis Our hypothesis is that our HI-MoCA will be clinically equivalent to the original MoCA in our cognitively intact patients (with or without severe hearing loss). MATERIALS AND METHODS Study Methodology HI-MoCA Test Development and Procedure. Permission was obtained from Dr. Ziad Nasreddine, MD, to modify the MoCA into an hearing-impaired MoCA (HI-MoCA). We also obtained institutional ethics approval to complete this study. To administer the HI-MoCA to subjects without any verbal instructions or prompts, we converted the original verbal instructions into a visual PowerPoint (Microsoft Corp.) presentation on a computer screen. The patients read the instructions, and after acknowledging their understanding of the instructions, press a button to advance the presentation. They are also given a series of blank answer sheets to be completed as instructed by the PowerPoint presentation. Once advanced, the instructions disappear and are replaced with instructions to complete the task on the sheet of paper in front of them. Once the task is completed, the completed answer sheet is corrected so answers cannot be changed after the task is completed. The next blank answer sheet then awaits the subject to complete while, the next task is explained visually on the computer screen. These visual instructions are sufficient for sections on the MoCA, such as the visuospatial/executive tests (trails, copy cube, and clock draw) (Fig. 1). Visual instructions also are used in the naming section (Fig. 2). In the memory recall section, a series of words are presented at a rate of 3 seconds per word (face, velvet, church, daisy, red), comparable to the standard verbal administration of the original MoCA. A blank answer sheet is used by the subject to recall these words in writing. Immediately following this, the completed answer sheet is taking away and subjects are directed to the next blank answer sheet for a second trial recall. For the attention task requiring the participant to identify the letter A among a string of letters by tapping the table, the HI-MoCA visually presents a series of letters at a rate of 2 seconds between letters for the subject to observe and respond to by tapping the table as well. Finally, in the abstraction subtest, the HI-MoCA test administrator has additional slides that can be used if required. For example, the subject is asked to write down the similarity between a banana and orange. The correct answer is “they are Lin et al.: Development of Hearing-Impaired MoCA Fig. 1. Screen capture of the visually described trails task for the Hearing-Impaired Montreal Cognitive Assessment, along with the corresponding patient worksheet [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.] both fruit.” However, if the subject instead writes that both can be eaten, the test administrator can advance to a screen that displays the statement: “The answer we were looking for is that an orange and a banana are both fruit,” similarly to the verbal cues provided in the original MoCA. Participants Using the original MoCA validation study20 as a guideline for sample sizes, 103 participants were prospectively recruited from our adult cochlear implant program. Group 1 consisted of 103 subjects with normal hearing and normal cognition. Group 2 consisted of 49 subjects with severe hearing loss and normal cognition. Participants were all over the age of 60 and were excluded if they had a family history of dementia or had a history of any neurological disease. A detailed personal health history was taken prior to enrollment in our study to ensure that patients, both normal hearing and hearing impaired, were as cognitively intact as possible. In addition, three screening questionnaires developed for identifying cognitive impairment were administered, and patients also were excluded if they failed any of these assessments. These three questionnaires were: 1. Personal Health Questionnaire, including hearing screening, Developed in-house at Sunnybrook Health Sciences Centre 2. Activities of Daily Living Questionnaire by proxy (completed by a spouse, partner, or caregiver)25 Fig. 2. Screen capture of the visually described animal-naming task for the Hearing-Impaired Montreal Cognitive Assessment, along with the corresponding patient worksheet [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.] Laryngoscope 127: 2017 Lin et al.: Development of Hearing-Impaired MoCA S7 Fig. 3. Testing timeline for groups 1 and 2. MoCA 5 Montreal Cognitive Assessment; HI-MoCA 5 Hearing-Impaired Montreal Cognitive Assessment. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.] 3. Alzheimer’s Society of Canada screening questionnaire for primary care physicians26 Based on prior clinical experiences, our intake screening, along with the three administered questionnaires, are effective in excluding a significant majority of patients with early cognitive impairment who previously have not been diagnosed. Subjects within group 1 (normal hearing, normal cognition) were randomized to receive either the standard MoCA (n 5 43) or HI-MoCA (n 5 60) during their first visit (T1). Approximately 6 to 8 months later (T2), subjects were retested with either the MoCA or HI-MoCA, which they did not receive during their first visit. This inter-test interval was selected to minimize any potential learning effects from repeating the MoCA (Fig. 3). Subjects in group 2 (severe hearing loss, normal cognition) were given the HI-MoCA test during their first visit and subsequent visit 6 to 8 months later. In total, eight versions of the HI-MoCA were evaluated. In the earlier versions, a small pilot group of different subjects (5–10) underwent testing and then interim analyses were performed, along with extensive discussions, among our research group. Next, the HI-MoCA test was modified to incorporate these changes. Many of the changes were made to help streamline the test administration and reduce the need for prompting or tester intervention. This final data set was collected using the eighth version of the HI-MoCA on subjects who were not involved in the prior pilot studies. Statistical Analyses Descriptive statistics, including averages and standard deviations (SD), were used to summarize the results of the respondents, their MoCA/HI-MoCA scores, and the time between tests in weeks. Prior to selecting the type of statistical test used, we tested for normality. Because our data was normally distributed, a paired t test was used to compare MoCA and HI-MoCA scores, as well as HI-MoCA scores between groups. Statistical significance was defined as a P < 0.05. When multiple comparisons were made in the MoCA subset analysis, the P value was adjusted to account for these multiple comparisons (0.05 divided by number of comparisons made). A two-way repeated measures analysis of variance (ANOVA) was run to determine whether the order of the testing—original MoCA (T1), then HI-MoCA (T2 vs. HI-MoCA [T1]), and then original MoCA (T2)—would impact the scoring for group 1. Analysis of the studentized residuals and the ShapiroWilk test were used to determine normality. Sphericity of the interaction term (order and time) was assessed by Mauchly’s test, and a priori it was determined that simple main effects would be run if the interaction term was statistically significant. Test–retest reliability was calculated using the standard coefficient when testing between time periods in group 2 for the HI-MoCA. RESULTS Results: Group 1 (Normal Hearing, Normal Cognition) and Group 2 (Severe Hearing Loss, Normal Cognition) There were 103 subjects in group 1 who passed the intake form and all three screening questionnaires, and who completed both the original MoCA and HI-MoCA. Of these, 43 subjects underwent the original MoCA, first followed by the HI-MoCA 6 months later; and 60 subjects underwent the HI-MoCA, first followed by the original MoCA 6 months later (Table I). There was not statistical difference between group 1 or group 2 with regard to age and years of education. In group 1, the average HI-MoCA score was 26.66 (range: 20–30, SD 5 2.542). The original MoCA score was 27.14 (range: 21–30, SD 5 2.005). The original MoCA scores were statistically significantly better than the HI-MoCA scores, t(102) 5 22.11, P 5 0.037. This TABLE I. Demographic Data. Male Female Years of Education Average Age Number of Weeks Between Tests Group 1 (n 5 103) 29 74 15.65 (SD 5 2.25) 68.41 (SD 5 6.17) 28.93 (SD 5 6.17) Group 2 23 26 15.04 (SD 5 2.40) 70.23 (SD 5 6.74) 29.11 (SD 5 5.97) (n 5 49) Laryngoscope 127: 2017 S8 Lin et al.: Development of Hearing-Impaired MoCA TABLE II. MoCA and HI-MoCA Subtest Scores. Original MoCA HI-MoCA P Value (sig < 0.007) Visuospatial/executive 4.69 (SD 5 0.58) 4.54 (SD 5 0.73) 0.054 Naming Memory 2.85 (SD 5 0.38) 3.74 (SD 5 1.15) 2.92 (SD 5 0.27) 4.09 (SD 5 1.14) 0.145 0.005 Group 1: Subtest Attention 5.77 (SD 5 0.51) 5.64 (SD 5 0.79) 0.144 Language Abstraction 2.42 (SD 5 0.69) 1.72 (SD 5 0.51) 2.08 (SD 5 0.87) 1.54 (SD 5 0.72) 0.001 0.026 Orientation 5.92 (SD 5 0.27) 5.82 (SD 5 0.49) 0.063 Significant results are bolded. MoCA 5 Montreal Cognitive Assessment; HI-MoCA 5 Hearing-Impaired Montreal Cognitive Assessment; SD 5 standard deviation. data suggests that the HI-MoCA is more challenging because subjects scored on average 0.48 points lower when compared to the MoCA. Exploratory follow-up analyses were conducted to determine whether this difference could be attributed to either one or several of the subtests (Table II). The 0.48-point difference between the HI-MoCA and MoCA scores was not thought to be clinically significant given that the scoring of both tests is based on whole numbers with the smallest incremental score of 1. If the difference was equal or greater than 1, it would have both statistical and clinical significance. We performed a post hoc sensitivity analysis. Using the current cutoff score of 26 or greater as being a cognitive normal result, 28 of the 103 subjects scored below 26 and would be considered as failing this screening test. If we added 0.5 to all their scores, the same 28 of the 103 subjects would have failed. If we added 1.0 to all of their scores, then eight of the 28 subjects would then be considered within normal range; thus, only 20 of the 103 subjects would fail the HI-MoCA screening test. Participants performed statistically significantly better on the memory section of the HI-MoCA (score: 3.74, SD 5 1.145) compared to the original MoCA, t(102)5 22.895, P 5 .005. (score: 4.09, SD 5 1.142). However, for the language subtests, the HI-MoCA score was statistically significantly lower, t(102) 5 3.57, P 5 .001 (score: 2.42, SD 5 0.69) than the original MoCA (score: 2.08, SD 0.087). All the subtest scores and P values are listed in Table II. A two-way repeated measures ANOVA was run to determine whether the order of the testing—original MoCA (T1), then HI-MoCA (T2 vs. HI-MoCA [T1)], and then original MoCA [T2]—would impact the scoring for group 1. Analysis of the studentized residuals showed that there was normality, as assessed by the ShapiroWilk test of normality with no outliers, as assessed by no studentized residuals greater than 6 3 SDs. There was sphericity for the interaction term, as assessed by Mauchly’s test of sphericity (P > .05). There was a statistically significant interaction between order and time on MoCA outcome, F(1, 101) 5 6.08, P 5 .015, partial g2 5 .057. Therefore, simple main effects were run. MoCA scores were statistically higher different if subjects Laryngoscope 127: 2017 received the HI-MoCA first and the original MoCA second, F(1,42) 5 11.695, P 5 .001, partial g2 5 .002, compared to if they received the original MoCA first and the HI-MoCA second, F (1,59) 5 .003, P - 0.96, partial g2 5 0. See Table II for means and SDs. There were 49 subjects in group 2 who passed all three screening tests and completed the HI-MoCA twice, approximately 6 months apart. In group 2, the average HI-MoCA score when tested for the first time (T1) was 26.184 (SD 5 2.579). Approximately 6 months later when re-tested (T2), the average HI-MoCA score was 26.493 (SD 5 2.639). There was no statistically significant difference between the HI-MoCA scores between the two time points, t(48) 5 2.995, P 5 0.324. The test–retest reliability coefficient was 0.66, indicating excellent test–retest reliability (Table 3). Results: Comparing HI-MoCA Scores in Normal Hearing (Group 1) and Severely HearingImpaired (Group 2) Groups We compared HI-MoCA scores in both group 1 and group 2 subjects, which then were further analyzed to determine whether severe hearing loss could impact HIMoCA scores. Group 1 subjects performed the HI-MoCA either at the initial test period (n 5 60) or at the 6month period (n 5 43). Their average HI-MoCA score was 26.66 (SD 5 2.642, standard error [SE] mean 5 0.378). Group 2 subjects were tested with the HI-MoCA twice. Because the test–retest reliability coefficient was very high (0.66) and a significant memory learning effect was ruled out, for this comparison we only used the HIMoCA score on the first test timepoint (T1). The HIMoCA score for group 2 subjects was 26.184 (SD 5 2.579, SE mean 5 0.368). There was no statistically significant difference in HI-MoCA scores between group 1 or group 2 subjects (t(48) 5 1.138, P 5 0.261). Further subtest analyses did not reveal any statistically significant differences in scores on the HI-MoCA between group 1 and group 2 subjects (Table 4). DISCUSSION The goal of the present study was to develop a visual, computer-administered version of the MoCA for use Lin et al.: Development of Hearing-Impaired MoCA S9 in hearing-impaired individuals to directly address the recognized need for tests adapted to individuals with sensory impairments.22,23 We believe that, with our results, our HI-MoCA is equivalent to the MoCA in our tested population. Study Design Metrics We recognize that the conversion of auditory instructions to visual instructions would involve different neural pathways. Although a detailed neuroanatomical discussion on the differences between auditory versus visual processing pathways is beyond the scope of this article, we strongly believed that our modifications were minimal but necessary to achieve our study objective. Even among experts within the field of neuroscience, there still is debate on the precise neural pathways involved with instruction-based task sets, irrespective of whether they are auditory or visual in nature.27 We also believed that although the neural entry point with auditory and visual instructions are unique, the pathways involved with processing the instructions, storing information, and ultimately performing the requested tasks all merge into common pathways. Furthermore, if changes were dramatic, differences in scoring would become apparent with the testing of our two distinct groups using these two versions of the MoCA. As demonstrated in our results, only small differences were noted, and none were clinically significant. We also considered how best to eliminate patients with any symptoms of cognitive decline for inclusion into either group 1 or 2. In the original validation article, Naasreddine et al.20 used a very comprehensive psychometric battery of tests for this determination. We initially considered performing concurrent formal neuropsychological testing, but this was not possible given that there are no versions that can be administered to the hearing impaired. Based on prior screening efforts by our group, a brief historical intake form and the three questionnaires was determined to be adequate. The inclusion of an activities of daily living questionnaire by proxy, which was completed by the caregiver, further decreased the likelihood that patients with significant cognitive impairment and functional decline would be recruited. The global MoCA and HI-MoCA results indicate that the majority of our subjects in this study are cognitively intact. statistically significant, it is not clinically relevant. Clinical relevance would be for statistically significant differences of 1 or greater. Our post hoc sensitivity analyses did not confirm a clinically significant change in the number of subjects who would fail the HI-MoCA screening test. For screening tests to be widely utilized and adopted, the test and its scoring system must be simple, quick, and reliable. Although our HI-MoCA is arguably more complex than the original MoCA, it remains an easy-to-administer and reliable cognitive screening test. As clinicians accustomed to working with the hearing impaired well know, any form of interaction necessitates an adapted means of communication, which is timeconsuming. Consideration of Learning Effect and Test– Retest Reliability We also considered the possibility of whether subjects would improve their MoCA scores when being tested for the second time. We decided that a 6-month window would be sufficiently long for subjects not to demonstrate any significant memory effects. We also were cognizant of the fact that a longer duration may increase the window in which new health issues change the subject’s cognitive state. Furthermore, a longer follow-up period also could result in a higher dropout rate among our subjects. Fortunately, in our group 2 (severely hearing impaired, normal cognition), the HI-MoCA scores at T1 and T2 were similar and not statistically significantly different (26.66 vs. 26.18, P 5 0.261). The test–retest reliability coefficient was 0.66. Had we encountered a memory learning effect in which the T2 scores were significantly improved, we would have used alternative versions of the MoCA to convert into several HI-MoCA versions as well. The original MoCA has a second and third version in which the subtests are different. For example, rather than identify the standard 3 animals, version 2 has a giraffe, bear, and rhinoceros. The word list, number sequence, language, and abstraction examples also are different. For simplicity, we elected to stay with one version. Consideration of Test Order Consideration of a Correction Factor In our validation study, we found that in group 1 subjects (normal hearing, normal cognition), there was a statistically significant difference in scores for the HIMoCA versus the MoCA (26.66 vs. 27.14). This difference in score was 0.48. Further subtest analysis revealed that two subtests of the seven were statistically significantly different between the two versions of the MoCA (memory and language). However, the actual mean subtest difference ranged between 0.35 and 0.34. We considered a correction factor to account for this difference. However, the MoCA is scored with whole numbers with the smallest incremental score of 1. Although a 0.48 difference in score between the HI-MoCA versus the MoCA might be Laryngoscope 127: 2017 S10 We also asked whether the first test, regardless of whether it was the HI-MoCA or MoCA, would factor in the subjects’ scores. This was analyzed using the group 1 subjects only because they received both tests. A twoway repeated measures ANOVA analysis of both sequences (MoCA [T1] and then HI-MoCA [T2] vs. HI-MoCA [T1] and then MoCA [T2]) did reveal that those in group 1 who were first evaluated with the HI-MoCA did worse on the HI-MoCA than those who were first tested with the MoCA. This difference was a challenge to fully understand. Those who were first tested with the HI-MoCA had an average score of 26.12 and then scored 27.23 on the original MoCA, for a difference of greater than 1 point. Lin et al.: Development of Hearing-Impaired MoCA When the sequence of tests was reversed, the participants scored 27.07 and then scored 27.05 on the HIMoCA. We also note that the group 2 subjects who had the HI-MoCA administered twice did not have any significant difference from T1 to T2 on their scores. Given that this improvement in scoring was only seen in this one instance, we decided it was not evidence of a learning effect. Again, this difference is not clinically significant because the clinical utility of this test is designed for a single time point and not meant to be repeated. Future Directions We also have begun to test our HI-MoCA on a group of subjects with clinically confirmed MCI. The purpose of this study is to ensure that our HI-MoCA test results also coincide with MoCA scores in patients who score predominantly below the cutoff range of 26 and who complete the necessary spectrum of patients of varying cognitive functioning to fully validate the HI-MoCA. With this set of data, we will be able to either confirm 26 or potentially a lower threshold in which patients would be sent for further cognitive evaluations. Our future direction also will include a smallerscale validation study using the original MoCA version 2 and 3, as well as a direct French HI-MoCA translation. We also are in consideration of converting the timed PowerPoint (Microsoft Corp., Redmond, WA) HI-MoCA presentation into a multi OS mobile application (iOS and Android). This mobile application, if run on a tablet, could serve as both the testing platform and the recording and scoring platform, and it may allow worldwide collaboration and the collection of data for future studies and projects. With the completion of this study, the HI-MoCA is being utilized at our tertiary adult cochlear implant clinic to screen-selected patients over the age of 60 years who may have signs of cognitive decline. We have also distributed this test to various centers and colleagues in North America, Europe, and Australia. CONCLUSION The HI-MoCA is an easy-to-administer and reliable screening test for cognitive impairment in the severely hearing impaired, with no conversion factor required in our tested cohort of cognitively intact subjects. Acknowledgment We wish to thank Dr. Tony Esklander and Dr. Jane Opie for assistance and advice on statistical analyses and article preparation. We would also like to thank Dr. Ziad Nasreddine MD for his permission to modify the MoCA to the HIMoCA for this project. Laryngoscope 127: 2017 BIBLIOGRAPHY 1. Uhlmann RF, Larson EB, Rees TS, Koepsell TD, Duckert LG. Relationship of hearing impairment to dementia and cognitive dysfunction in older adults. 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