Patient-Reported Satisfaction and Associated Changes in Health and Behavior Pre- and Post- Project INSPIRE: a Comprehensive Hepatitis C Care Coordination Program

Individuals infected with hepatitis C (HCV) often present with co-morbidities and complex socio-behavioral risk factors. Project INSPIRE was a care coordination and telementoring demonstration project which aimed to treat and cure HCV-infected patients while providing them with services and education to improve overall health outcomes and self-suciency. We examined changes in HCV-related health and behavior associated with completion of Project INSPIRE (the “intervention”). Methods to patients, providers, and insurance companies beyond the immediate benets of HCV treatment and cure. Participants also had a generally positive experience working with their care teams, indicating that further research should explore how an individualized care team can improve retention and patient referral rates compared with the HCV standard of care. the multivariable models assessing various self-reported health behaviors toward INSPIRE’s achieving other program goals. Completion of INSPIRE associated with signicant reductions in self-reported ED visits, hospitalizations, alcohol use, and injection drug use. Increased self-ecacy between enrollment and completion of INSPIRE suggests achievement of one of INSPIRE’s main program goals: improving participants’ ability to manage their own health and navigate the healthcare system.


Introduction
Hepatitis C virus (HCV) causes an infection that, when left untreated, can lead to liver damage, including cirrhosis, hepatocellular carcinoma, and liver failure (1). Currently, an estimated 2.4 million persons (1% of the population) in the United States (US) (2) and approximately 116,00 persons in New York City (NYC) (3) are chronically infected with HCV. People most at risk for infection include injection drug users, those currently and formerly incarcerated, and those in the "baby boomer" birth cohort (individuals born between 1945 and 1965) (4).
to receive a post-intervention survey within one month of their last visit with their care coordinator regardless of whether they had completed a baseline survey.

Survey and data collection
Each survey packet included an information ier, the survey in English (6 pages) or Spanish (8 pages), an informed consent form, and a stamped, pre-addressed envelope to return the survey to New York City Department of Health and Mental Hygiene (NYC DOHMH). All materials were available in English and Spanish and distributed according to patients' language preference. Surveys and informed consent forms were to be completed by participants without the assistance of their care coordinator or clinician (the care team). Respondents were directed to contact the NYC DOHMH evaluation team using the phone number on the information ier with questions or for clari cation in order to limit care team involvement. Participants also had the option to call the evaluation team and complete the survey over the phone. For each returned baseline or post-intervention survey, respondents were mailed a $25 gift card. Respondents were contacted by phone if response clari cation was necessary or questions were unanswered. All survey packets contained the same information and all surveys asked the same questions, although during the rst few weeks of survey deployment a few minor changes were made to the survey itself. These included adding graphics to the cover page to make it more appealing, changing the responses from ll in the blank to categorical for number of ED visits and hospital nights in the past 6 months and removing the patient satisfaction questions from the baseline survey since the questions were only intended for post survey respondents.
Secondary data source INSPIRE clinical sites submitted program data to NYC DOHMH in a clinical database which included participant name, contact information, sociodemographic information, dates of enrollment, care coordination services, treatment initiation and completion, discharge and SVR/cure status. First and last name of respondents and enrollment site listed on the returned surveys were used to identify respondents in the clinical database to link the two datasets.

Measures of interest
The main goal of the analysis was to assess changes in overall self-reported health status and health-related behavior, HCV-related knowledge, and self-e cacy in regard to managing health between baseline and postintervention. Seven outcomes of interest were collected in both the baseline and the post-intervention survey: selfreported overall health (measured using the single general self-rated health question (GSRH) (15)) (outcome 1); self-reported number of emergency department (ED) visits (outcome 2), number of nights hospitalized (outcome 3), any drug use (outcome 4), and any alcohol consumption (outcome 5) in the past 6 months; HCV knowledge (assessed using a 14-question HCV knowledge scale adapted from the validated Brief HCV Knowledge Scale) (16) (outcome 6); and respondent self-e cacy (assessed using a 13-question self-e cacy measure adapted from the validated Patient Activation Measure) (17). The self-e cacy measure was su ciently different from the original Patient Activation Measure, so the original scoring guide could not be used to score the adapted measure. Exploratory factor analysis (EFA) and subsequent factor scoring was instead used to generate respondent scores (outcome 7). Both surveys also included questions on demographics, speci cally education and marital status.
Secondary goals were to measure patient adherence to their HCV medication and patient satisfaction with their HCV care coordinator, HCV medical provider, and the INSPIRE health promotion sessions at the end of the intervention. HCV medication adherence was assessed via two adapted questions (18) asking about the respondent's adherence during the preceding four weeks. Respondent satisfaction with their care coordinator and clinician was measured with questions from the Patient Reactions Assessment (19) regarding quality of information received, perceived quality of communication, perceived affection, and perceived respect. Satisfaction with health promotion sessions was assessed using ten questions developed by the INSPIRE evaluation team. Responses were recoded into Agree (agree + strongly agree) and Neutral/Disagree (neutral + disagree + strongly disagree), in keeping with patient satisfaction analysis convention (12). Responses of "not applicable" were coded as missing.

Factor analysis
To generate self-e cacy scores, we used EFA to identify latent constructs ("factors") underlying responses to the self-e cacy questions in the survey and to score responses based on this factor structure (20). The data were checked and met the assumptions for EFA analysis (21)(22)(23)(24)(25). Factors were then extracted using principal axis factoring and oblique rotation (24), taking into account non-normal distribution of responses (26) and communalities (multiple questions de ning the same factor) (20,25). A one-factor structure was hypothesized, based on prior assessment of the unmodi ed Patient Activation Measure (17) and con rmed based on factor retention guidelines (24,25). Each patient's self-e cacy question responses were then scored and standardized based on this one-factor structure. To account for the subjective methodologies used to determine factor retention (27) and compute factor scores (20), a sensitivity analysis was performed using respondents' mean self-e cacy score generated from the original Patient Activation measure scoring guide in place of factor score.
Crude and adjusted generalized estimating equation (GEE) models for a respondent's mean self-e cacy score were calculated and the results were compared with models using factor scores.

Response rates
Survey responses were classi ed as complete, partial, break-off, ineligible, and lack of response ("refusal"), per American Association for Public Opinion Research (AAPOR) guidance (28). Surveys with responses for > 80% of the questions were counted as complete, those with responses for 50-80% of the questions were counted as partial, and those with responses for < 50% of the questions were deemed too incomplete to process (break-off, ineligible, or lack of response) and subsequently excluded from analyses. Response rates were calculated using the AAPOR de nition for mail surveys of speci cally named persons (all surveys were returned via mail except for 2 conducted over the phone), both for only complete surveys and for complete and partial surveys. The AAPOR publicly available Survey Outcome Rate Calculator version 4.0 (mail_SN) was used to conduct the calculations (29).

Power limitations
Baseline and post-intervention surveys received from the same respondent were classi ed as "paired"; when only the baseline or post-intervention survey was available, it was classi ed as "unpaired." Because of the limited number of paired surveys available, we included all eligible surveys. In order to maximize power, we avoided sub-analyses because of small numbers, and we only used covariates shown to be important for the outcome in existing literature rather than based on statistical signi cance (13,30).

Summary analyses
Descriptive statistics, including statistical tests of association, were used to compare sociodemographic characteristics between individuals with paired and unpaired surveys and between baseline and post-intervention survey respondents within the unpaired group. We evaluated differences between (paired vs unpaired) and within groups (unpaired baseline vs unpaired post) to determine what results in subsequent analyses we could attribute to the effect of INSPIRE versus differences in response rates (unpaired group). Wilcoxon-Mann Whitney tests were used to assess continuous variables, and chi-square tests were used to assess categorical variables. By comparing the between and within groups together, rather than directly baseline and post, all observations were independent and we did not need to account for the overlapping nature of the sample. Prior to analyses, values of "unknown" and "refused" were coded as missing and excluded from all summary analyses. Following descriptive analyses, missing values for survey variables were imputed (see Missing Data below). Differences in HCV knowledge were assessed by comparing the percentage of correct responses among baseline and postintervention questions using chi-square tests that accounted for the overlapping sample (31). For the summary analyses, data about alcohol and IDU use in the last 6 months were collected at time of enrollment during the comprehensive assessment while for the outcome analyses survey responses were used.

Missing data
To include all eligible surveys in the multivariable analyses, multiple imputation (MI) was used to preserve surveys with missing responses. Given that social desirability bias likely in uenced the response rates to some questions, we assumed that the data were missing not at random (MNAR). However, we chose to proceed with MI because the technique can still produce unbiased results under the MNAR mechanism (32). After assessing associations between various demographic or survey variables and survey completeness using t-and chi-square signi cance tests, the nal variables included in the imputation process were survey type, age, gender, race/ethnicity, marital status, social support, HIV status, mental health status, and all survey variables assessed as one of the seven primary outcomes speci ed above, except for self-e cacy. Missing values for the selfe cacy questions were not imputed to simplify subsequent EFA.
MI with chained equations, generating 20 imputed datasets, was performed (33) using all variables included as predictors for imputation for each variable with missing values and using marital status and survey type as auxiliary variables. MI was performed using the IVEware stand-alone SRCWare package (34). To account for the MI, PROC MIANALYZE was used in the outcome analyses to generate a single estimate for each regression coe cient by synthesizing the values from all imputed datasets.

Outcome analyses
The changes in the following outcomes before and after receiving the INSPIRE intervention were assessed using GEE regression models: (1) overall health, (2) number of ED visits in the preceding 6 months, (3) number of nights spent in the hospital in the preceding 6 months, (4) any alcohol and (5) any drug use in the preceding 6 months, (6) HCV knowledge, and (7) self-e cacy. The self-reported health outcomes (1-3) were analyzed as ordinal multinomial variables, while the self-reported behavioral variables (4 and 5) were analyzed as binary variables. The HCV knowledge score (range: 0-100%) and self-e cacy scores (range: 0-5) were analyzed as continuous variables (6 and 7). GEE regression models, with exchangeable correlation structure, were used because of the non-independence of the paired baseline and post-intervention survey responses, and the non-normal distributions of some variables [35][36][37][38]. To account for the exchangeable correlation structure for binary and ordinal outcomes, the alternating logistic regression (ALR) method was used in the models for these outcomes. The ALR approach uses odds ratios (ORs) to model within-respondent associations and calculates the odds of a change in the response to a given measure in the post-intervention survey relative to the baseline survey (35).
The initial confounders used in all adjusted models were age at enrollment, gender, race/ethnicity, education level, social support, HIV status, and mental health status, selected based on preexisting literature (13,30) since not all variables included in the summary analyses were included in the model due to over dispersion. To account for model non-convergence, variables found to not contribute to the model based on the literature, followed by testing the statistical signi cance with the outcome, were removed (36). Race/ethnicity was removed from the alcohol use model, and race/ethnicity and education level were removed from the drug use model. Tests of association showed that respondents with paired and unpaired surveys, as well as respondents returning only either baseline or post-intervention surveys were mostly similar for almost all demographic characteristics. The only statistically signi cant difference was that among respondents with unpaired surveys, post-intervention-only survey respondents tended to be older than baseline-only survey respondents (mean age 59.4 years compared with 53.8 years, respectively, p = 0.0004) ( Table 1). Although we might have been underpowered to detect differences in demographic, clinical, and sociobehavioral characteristics between groups of respondents who submitted only a baseline or only a post-intervention survey, we considered these groups similar enough to mimic paired surveys and merit inclusion in the GEE models. Prior to conducting multiple imputation, chi-square tests were conducted to compare the distribution of missingness between the complete and partial surveys for each of the analysis and auxillary variables used in the multiple imputation. Signi cant differences were found for education level (p = 0.01) and marital status (p = 0.004) ( Table 2). Overall, respondent satisfaction with their INSPIRE care coordinators, clinicians, and the health promotion sessions was very high (Fig. 2)-more than 80% of the subset of participants responding to the post-intervention survey selected "Agree" or "Strongly Agree" with positive statements regarding these aspects of the INSPIRE intervention.
Respondents tended to report very high medication adherence-more than 80% of respondents reported missing one dose of HCV medication or fewer in the preceding four weeks of treatment, and more than 80% reported following their treatment time schedule every day.

Changes in Health Behaviors and Self-E cacy, Baseline to Post-Intervention
Completion of INSPIRE was not associated with a change in HCV knowledge. Compared with an average baseline score of 82.4%, the average post-intervention score was 75.3%; however, after adjustment in the GEE model, the decrease was non-signi cant (adjusted average decrease: -4.01%; CI -9.10-1.08%; p = 0.12). Examined individually, two knowledge questions showed signi cant response changes, including if coughing and sneezing can spread HCV, and if a person can be re-infected with HCV after being cured. Although both questions showed, against expectation, statistically signi cant decreases in HCV knowledge, these results are not surprising given the high level of HCV knowledge at baseline (Table 3). showed a decrease in the odds of (across categories) ER visits (aOR = 0.34, 95% CI: 0.20-0.57) and a decrease in the odds of (across categories) hospital visits (aOR = 0.30, 95% CI: 0.16-0.57) compared with when participants were rst enrolled in INSPIRE. A decrease in alcohol and drug use (over the past 6 months) was associated with having received the INSPIRE intervention (aOR = 0.35, 95% CI: 0.18-0.6 9and aOR = 0.03, 95% CI: 0.03-0.34).
Given a maximum measure score of 5, this change represents a 6.8% increase (CI 1.6-12.2%) in the average score. Results from the sensitivity analysis using respondents' mean self-e cacy score instead of the factor score also showed a positive association between INSPIRE and reported self-e cacy with a 0.23-point (CI 0.05-0.41) increase (4.6%; CI 1.0-8.2%) (p = 0.01).

Discussion
This analysis indicates a positive association between participation in Project INSPIRE and improvement in selfreported health outcomes. Survey respondents, as with INSPIRE participants generally, were high-need-the majority were low-income, had less than a high-school education, experienced numerous comorbidities, and grappled with both substance use and mental health issues (unpublished data, NYC DOHMH). INSPIRE was wellliked and well-received by respondents and successful in improving HCV-related health behavior including reducing ED visits, hospitalizations, alcohol use, and drug use. Post-intervention survey respondents showed high satisfaction with INSPIRE, consistently agreeing with positive statements and disagreeing with negative statements about their care coordinator, clinician, and health promotion sessions. However, social desirability bias might have in uenced some participants to answer more favorably-although it was explained to participants that their responses would not be viewed by their care team, they may not have fully realized this, especially given that their care coordinator distributed the survey. However, the overall high level of satisfaction does suggest that the INSPIRE intervention was well-received by the participants who completed the survey. This also suggests that intervention recruitment might have bene ted from referrals from satis ed patients to their HCV-positive peers.
The health promotion sessions generated more mixed satisfaction responses than other project aspectsapproximately 70% of respondents "disagreed" or "strongly disagreed" with negative statements compared with approximately 80% of respondents when evaluating their care coordinators and HCV medical providers. These ndings were consistent with results from qualitative focus groups conducted to evaluate the INSPIRE model (unpublished data, NYC DOHMH). Participants from the focus groups indicated that they were sometimes dissatis ed with the quantity and length of sessions. The INSPIRE protocol was written to deliver 7 health promotion sessions, each scheduled to last 30 minutes, using content not necessarily tailored to each participant's knowledge or health literacy level. Future iterations of the program might require changes to the health promotion sessions to improve satisfaction.
Although INSPIRE aimed to improve participant HCV knowledge, the current analysis suggests this was not achieved. The lack of positive change might be because of a high average level of knowledge among the baseline group (83.3% on the HCV Knowledge Scale). With regression toward the mean, statistically signi cant increases in knowledge would be di cult to observe when the baseline score is already quite high (16).
Regardless of initial knowledge level, lack of signi cant improvement suggests a need for improved HCV messaging in the program. In addition, previous research has described that HCV infected individuals can develop cognitive impairment, directly due to the virus (37) and/or their potentially past use of alcohol and drugs. It may thus be important to account for this in the design of health promotion modules so that content is delivered at an appropriate level and at multiple time points to maximize participant comprehension and retention. improving participants' ability to manage their own health and navigate the healthcare system.
As a demonstration project, INSPIRE was intended to evaluate the effect of providing tailored care coordination on treatment outcomes for HCV patients. This study shows that the advantages of INSPIRE might go beyond just clinical outcomes such as treatment initiation and SVR. These ndings suggest that improvements in speci c health behaviors and perceived self-e cacy could be expected after the participation in and completion of Project INSPIRE. These improved longer-term outcomes could be of interest to groups such as insurance companies and managed care organizations that might be concerned about the upfront added costs of care coordination but are interested in the possible savings from decreased ED visits, inpatient hospitalizations, and improved self-e cacy, which has been linked to better primary care outcomes and reduced costs (38).
There are some limitations associated with this study. Non-response and selection bias likely played a large role in these results. The response rates for the baseline and post-intervention survey were very low-14.0% and 9.9%, respectively, for all returned surveys-particularly compared with the > 40% response rate for mailed surveys reported in other studies (39). The low response rates limit our ability to generalize the results of this study to the entire INSPIRE cohort. Also, the majority of returned surveys were unpaired, limiting our ability to draw conclusions about changes in individuals.
Although edits to the survey were minimal, initial iterations might have been less appealing to complete, thus in uencing who responded and how they answered the survey questions. Survey distribution was not uniform across care coordinators-although the survey protocol stated all eligible participants should receive the survey, no formal process ensured care coordinators distributed the survey as instructed with the same level of information, such as whom to contact for questions, purpose of the survey or possibly providing assistance completing the survey. Furthermore, we were not able to perform an assessment of response bias comparing survey respondents with the entire INSPIRE cohort because of IRB restrictions.
Care coordinators and respondents highlighted further limitations of the survey. These issues included a survey literacy level higher than that of some of the respondents, the length of the survey, and confusion around the wording of some questions. While the research team sought to answer and follow up on any respondent questions, we were not able to contact all respondents who potentially had misunderstandings. This might have created variability in response, biasing results. Confusion might also have been caused by a simultaneous ongoing survey distributed by outside INSPIRE evaluators hired by the funder, possibly causing participants to believe this survey was a duplicate of a previously completed survey and contributing to a lower response rate.
Despite these limitations, the ndings from this study provide insights into INSPIRE participants' perspectives on HCV-related care coordination services. Based on the analyses presented here, the program successfully improved respondent health-related behaviors, including self-perceived ability to manage their own health, and these results complement past ndings that suggest the intervention is worth replicating in the future, including with other chronic medical conditions. Noted improvements in this complex population also highlight the positive impact this model may have in populations facing similar barriers to care and should be considered in future public health intervention planning for persons with chronic illness. Further research will be key to understanding which components of INSPIRE are the most effective at improving clinical, behavioral, and overall health outcomes and how to address possible areas for program improvement.