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Effect of Behavioral Cough Suppression Therapy Delivered via Telehealth

Published:December 20, 2022DOI:https://doi.org/10.1016/j.jvoice.2022.11.015

      Summary

      Objectives/Hypothesis

      Behavioral cough suppression therapy (BCST) has demonstrated up to 88% effectiveness at treating refractory chronic cough (RCC). With onset of the COVID-19 pandemic, along with many other medical services, BCST shifted to telehealth delivery. Our group hypothesized that BCST delivered via telemedicine by a specialized Speech-Language Pathologist would be comparable to previously reported response to treatment for in-person settings.

      Study Design

      Retrospective review.

      Methods

      An Emory IRB approved, retrospective review of electronic medical records was completed for RCC patients who received BCST via telehealth from March 2020 through January 2022 at Emory Voice Center. Patients were included in the study if they had a diagnosis of RCC, were referred for BCST, were seen for at least one therapy session in the telehealth setting, and provided Cough Severity Index (CSI) data pre and post-treatment. Patients were excluded if they had incomplete datasets, a known pulmonary condition, structural laryngeal disorders, smoking history, dysphagia, and ACE-inhibitor use. Change in CSI score pre- and post-treatment was calculated to determine treatment effect. Paired-samples t-tests were conducted to compare pre-and post-treatment CSI score change.

      Results

      Fifty-one RCC patients were included in this study; 88% were female with an average age of 60 years (SD = 12.68). Post-treatment CSI scores were significantly lower than pretreatment CSI scores (P < 0.0001). These findings are comparable to historical documented CSI change achieved with in-person BCST.

      Conclusions

      This study provides preliminary evidence of the efficacy of BCST via telehealth for treating RCC. The findings of this study support the continued flexibility in speech-language pathology service delivery to include in-person and telehealth platforms for RCC beyond the COVID-19 pandemic.

      Key Words

      INTRODUCTION

      Behavioral cough suppression therapy (BCST) is a low-cost, low-risk, non-pharmacological treatment for patients living with refractory chronic cough (RCC). It is delivered by voice and upper-airway specialized speech-language pathologists (SLPs). BCST improves cough-related quality of life and reduces cough severity and frequency in up to 88% of patients with RCC after just 1 to 4 weekly sessions.
      • Vertigan AE
      • Theodoros DG
      • Gibson PG
      • et al.
      Efficacy of speech pathology management for chronic cough: a randomised placebo controlled trial of treatment efficacy.
      • Vertigan AE
      • Haines J
      • Slovarp L.
      An update on speech pathology management of chronic refractory cough.
      • Morice AH
      • Millqvist E
      • Bieksiene K
      • et al.
      ERS guidelines on the diagnosis and treatment of chronic cough in adults and children.
      • Ryan NM
      • Vertigan AE
      • Gibson PG.
      Chronic cough and laryngeal dysfunction improve with specific treatment of cough and paradoxical vocal fold movement.
      • Ryan NM
      • Vertigan AE
      • Bone S
      • et al.
      Cough reflex sensitivity improves with speech language pathology management of refractory chronic cough.
      • Chamberlain Mitchell SAF
      • Ellis J
      • Ludlow S
      • et al.
      Non-pharmacological interventions for chronic cough: The past, present and future.
      • Slovarp L
      • Jette M
      • Gillespie A
      • et al.
      Evaluation and management outcomes and burdens in patients with refractory chronic cough referred for behavioral cough suppression therapy.
      During the COVID-19 pandemic, many clinical settings rapidly incorporated telehealth (ie, virtual delivery of healthcare services via videoconferencing platforms) in lieu of in-person visits.
      • Anthony B.
      Use of telemedicine and virtual care for remote treatment in response to COVID-19 pandemic.
      • Monaghesh E
      • Hajizadeh A.
      The role of telehealth during COVID-19 outbreak: a systematic review based on current evidence.
      • Portnoy J
      • Waller M
      • Elliott T.
      Telemedicine in the Era of COVID-19.
      U.S. Department of Health and Human Services
      Medicare payment policies during COVID-19.
      Given the risk of airborne transmission of the virus,
      World Health Organization
      Transmission of SARS-CoV-2: Implications for Infection Prevention Precautions.
      it was imperative that SLPs pivoted from traditional models of care to a safer virtual option.
      • Doll EJ
      • Braden MN
      • Thibeault SL.
      COVID-19 and speech-language pathology clinical practice of voice and upper airway disorders.
      American Speech-Language-Hearing Association
      Considerations when providing voice services in the absence of endoscopic evaluation during COVID-19.
      • Castillo-Allendes A
      • Contreras-Ruston F
      • Cantor-Cutiva LC
      • et al.
      Voice therapy in the context of the COVID-19 pandemic: guidelines for clinical practice.
      The American Speech-Language-Hearing Association (ASHA) specifies that telehealth must not compromise quality when compared to in-person services.
      American Speech-Language-Hearing Association
      Code of Ethics.
      Since emergency exceptions were in place during the height of the COVID-19 pandemic to allow for rapid expansion of telehealth,
      U.S. Department of Health and Human Services
      Medicare payment policies during COVID-19.
      a unique opportunity was presented to examine the efficacy of telehealth interventions for SLP services, including RCC.
      Telehealth was viewed positively by Americans prior to the COVID-19 pandemic for reasons such as convenience and cost savings,
      American Well
      Telehealth Index: 2019 Consumer Survey.
      and there are many potential benefits of maintaining more flexible service delivery models in the SLP field. Telehealth delivery of voice therapy for a variety of conditions has been shown to be feasible, appropriate, and as efficacious as in-person therapy.
      • Doll EJ
      • Braden MN
      • Thibeault SL.
      COVID-19 and speech-language pathology clinical practice of voice and upper airway disorders.
      ,
      • Fu S
      • Theodoros DG
      • Ward EC.
      Delivery of intensive voice therapy for vocal fold dodules via telepractice: a pilot feasibility and efficacy study.
      • Kelchner L.
      Telehealth and the treatment of voice disorders: a discussion regarding evidence.
      • Mashima PA
      • Birkmire-Peters DP
      • Syms MJ
      • et al.
      Telehealth: voice therapy using telecommunications technology.
      • Theodoros DG
      • Hill AJ
      • Russell TG.
      Clinical and quality of life outcomes of speech treatment for Parkinson's disease delivered to the home via telerehabilitation: a noninferiority randomized controlled trial.
      • Rangarathnam B
      • McCullough GH
      • Pickett H
      • et al.
      Telepractice versus in-person delivery of voice therapy for primary muscle tension dysphonia.
      • Lin F-C
      • Chien H-Y
      • Chen SH
      • et al.
      Voice therapy for benign voice disorders in the elderly: a randomized controlled trial comparing telepractice and conventional face-to-face therapy.
      • Kuruvilla M
      • Hatcher J
      • Shelly S
      • et al.
      Virtual interprofessional chronic cough clinic: an efficient and appealing approach to a complex problem.
      A recent study explored the merits of a virtual interprofessional cough clinic and researchers found this model was highly satisfactory for RCC patients and efficient for clinical providers.
      • Kuruvilla M
      • Hatcher J
      • Shelly S
      • et al.
      Virtual interprofessional chronic cough clinic: an efficient and appealing approach to a complex problem.
      The telehealth model also addresses a shortage of specialized SLPs in rural areas across the country
      • Mashima PA
      • Doarn CR.
      Overview of telehealth activities in speech-language pathology.
      by connecting SLPs to patients who perhaps would not otherwise have the resources to access specialty care.
      • Towey MP.
      Speech therapy telepractice for vocal cord dysfunction (VCD): MaineCare (Medicaid) cost savings.
      To the best of our knowledge, only one other study based in the U.K. investigated the efficacy of Skype delivery of BCST to patients with RCC. Lillie et al. (2014) reported a 6-month case series of 11 patients with RCC and a related condition, vocal cord dysfunction/inducible laryngeal obstruction (VCD/ILO), who received SLP services via Skype. Endoscopic assessment was completed in person prior to initiating telehealth services where patients participated in a minimum of four sessions. Specific treatment activities were not described. Cough-specific quality of life improved by a clinically significant degree (minimal clinically important difference [MCID] = 1.3
      • Raj AA
      • Pavord DI
      • Birring SS.
      Clinical cough IV: what is the minimal important difference for the Leicester Cough Questionnaire?.
      • Birring SS
      • Prudon B
      • Carr AJ
      • et al.
      Development of a symptom specific health status measure for patients with chronic cough: Leicester Cough Questionnaire (LCQ).
      ) with an LCQ median score change of 5.8 and with all patients reporting being at least “very satisfied” with services received.
      • Lillie S
      • Haines J
      • Vyas A
      • et al.
      P112 Speech and language therapy by Skype for vocal cord dysfunction and chronic cough.
      The purpose of the present study was to evaluate the effectiveness of telehealth delivery of BCST at a university clinic during the COVID-19 pandemic. We hypothesized that BCST delivered via telehealth would significantly improve cough-related quality of life. A secondary hypothesis was that cough improvements would be equivalent to those reported in the literature for patients who received in-person BCST. If these hypotheses were supported, the current investigation would contribute additional evidence to support a call for continued flexibility in SLP service delivery models to include in-person and telehealth platforms beyond the COVID-19 pandemic.

      METHODS

      A retrospective review of electronic medical records (EMR) of patients who received BCST via telehealth for RCC at an urban voice, swallowing and upper airway center (Emory Voice Center (EVC)) was completed. This review was approved by the Emory's Institutional Review Board (IRB00054821). Telehealth visits were synchronous, two-way audiovisual encounters conducted via a healthcare Zoom account with a voice and upper airway-specialized SLP. All patients received treatment by one of five specialized SLP's at the EVC. BCST of this included cohort received treatment to laryngeal efficiency in voice and breathing as well as emphasis on awareness of urge-to-cough (UTC) and cough suppression techniques in response to UTC.
      • Yang J
      • Crawley B
      • Dehom S
      • et al.
      Chronic refractory cough: objective improvement with aerodynamic-focused.
      UTC awareness training included identification of cough triggers, patient's perception of the amount time between UTC and cough onset, and successes and failure of avoidance strategies. Patients were also instructed in a variety of cough suppression techniques, including semi-occluded vocal tract postures during respiration and phonation, pursed lips breathing, effortful swallow, and chin-tuck swallow. The patients were instructed to use whatever techniques were most effective for them when UTC was identified. They were also instructed to practice these strategies a-symptomatically. Treatment also included individualized instruction in resonant voice in patients who also presented with muscle tension dysphonia, as is common in this population.
      • Vertigan AE
      • Theodoros DG
      • Gibson PG
      • et al.
      Efficacy of speech pathology management for chronic cough: a randomised placebo controlled trial of treatment efficacy.
      • Vertigan AE
      • Haines J
      • Slovarp L.
      An update on speech pathology management of chronic refractory cough.
      ,
      • Ryan NM
      • Vertigan AE
      • Gibson PG.
      Chronic cough and laryngeal dysfunction improve with specific treatment of cough and paradoxical vocal fold movement.
      • Ryan NM
      • Vertigan AE
      • Bone S
      • et al.
      Cough reflex sensitivity improves with speech language pathology management of refractory chronic cough.
      Cough Severity Index (CSI) were gathered from patients who participated in telehealth BCST pre and post-treatment from March 2020 to January 2022. The CSI is a valid and reliable cough-specific quality-of-life questionnaire with 10 items rated on a 5-point Likert scale. The total score ranges from zero to 40 with a lower score indicating improved quality-of-life. There is no established minimal clinically important difference (MCID) for the CSI, but a total score greater than 3.23 is considered “symptomatic for cough” (p.1933).
      • Shembel AC
      • Rosen CA
      • Zullo TG
      • et al.
      Development and validation of the cough severity index: a severity index for chronic cough related to the upper airway.
      All chart entries in the EMR that were coded both for chronic cough (R05) and CPT codes for SLP evaluation and treatment in the telehealth setting (ie, 92524 and 92507-95) were pulled for inclusion evaluation. A total of 940 chart entries were reviewed and aggregated, representative of 344 individual patients.
      Inclusion criteria were: diagnosis of RCC, referred for BCST, received at least one therapy session via telehealth, and completed CSI data pre-post treatment.
      Exclusion criteria were: patients with incomplete clinical data, known pulmonary conditions, structural laryngeal disorders, smoking history, dysphagia, and ACE-inhibitor use. Two hundred ninety-three patients of the 344-patient data set were excluded from the study, with the greatest contribution being incomplete or unreported baseline or post-BCST CSI scores. A total of 51 patients met inclusion criteria. This data set could not be compared to in-person BCST because baseline and post-treatment CSI scores were not routinely collected or recorded until 2020, at which time in-person BCST was no longer provided secondary to concerns for aerosolized disease-containing respiratory particles associated with the COVID-19 pandemic.
      Statistical analysis was conducted using SAS 9.4 software (SAS, Cary, NC). The primary outcome of interest was change in CSI scores, tested with a student's paired T-test.

      RESULTS

      Clinical (pre- and post-CSI) scores and demographic data were extracted from 51 patient charts. The majority of patients were female (N = 45; 88.24%) and had an average age of 60 years (SD = 12.68) (Table 1). Paired T-test revealed significantly decreased (improved) pre to post-treatment CSI scores (mean = -4.5490; 90% CI = -6.25, -2.8468; P < 0.0001). Table 2 presents the mean pre- and post- CSI scores stratified by sex.
      TABLE 1Patient Demographics and CSI Score Characteristics
      Demographics (N = 51)
      Age (Mean, SD, in years) Range60 (12.68) 28-85
      Sex
       Male6 (11.76%)
       Female45 (88.24%)
      Ethnicity
       African American14 (27.45%)
       Asian1 (1.96%)
       Caucasian30 (58.82%)
       Unreported6 (11.76%)
      CSI score (Mean, SD)
       Pre20. 98 (8.73)
       Post16.43 (10.36)
      TABLE 2Pre- and Post-treatment CSI Scores by Sex
      NPretreatment CSI scores Mean (SD)Post-treatment CSI scores Mean (SD)
      Female4522.28 (8.08)17.08 (9.94)
      Male611.16 (7.57)11.5 (13.02)

      DISCUSSION

      The findings of this study provide evidence that BCST delivered via telehealth is effective in treating RCC, based on significant CSI change pre to post-treatment. While efficacy of in-person delivery of BCST has been shown in several studies,
      • Vertigan AE
      • Theodoros DG
      • Gibson PG
      • et al.
      Efficacy of speech pathology management for chronic cough: a randomised placebo controlled trial of treatment efficacy.
      ,
      • Ryan NM
      • Vertigan AE
      • Bone S
      • et al.
      Cough reflex sensitivity improves with speech language pathology management of refractory chronic cough.
      ,
      • Slovarp L
      • Jette M
      • Gillespie A
      • et al.
      Evaluation and management outcomes and burdens in patients with refractory chronic cough referred for behavioral cough suppression therapy.
      ,
      • Chamberlain Mitchell SAF
      • Garrod R
      • Clark L
      • et al.
      Physiotherapy, and speech and language intervention for patients with refractory chronic cough: a multicenter randomized controlled trial.
      telehealth delivery of BCST has not been extensively studied. Telehealth is convenient, time and cost efficient.
      • Portnoy J
      • Waller M
      • Elliott T.
      Telemedicine in the Era of COVID-19.
      ,
      American Well
      Telehealth Index: 2019 Consumer Survey.
      Data show that treatment attrition is reduced and visit attendance improved when patients are given the option to receive telehealth services compared to in-person treatment.

      Cutchin GM, Shelly S, Petty B, et al. A comparison of voice therapy attendance rates between in-person and telepractice. Accepted for publication in. American Journal of Speech-Language Pathology.

      Results of the current study align with the existing literature on efficacy of RCC treatment, both pharmacologic and behavioral. Specifically, five studies evaluating the efficacy of medical and procedural therapies to treat RCC have used the CSI as a primary outcome measure.
      • Dion GR
      • Teng SE
      • Achlatis E
      • et al.
      Treatment of neurogenic cough with tramadol: a pilot study.
      • Zalvan C
      • Berzofsky C
      • Tiwari R
      • et al.
      Trigger reduction prior to systemic drugs for neurogenic chronic cough.
      • Simpson CB
      • Tibbetts KM
      • Loochtan MJ
      • et al.
      Treatment of chronic neurogenic cough with in-office superior laryngeal nerve block.
      • Dhillon VK.
      Superior laryngeal nerve block for neurogenic cough: a case series.
      • Litts JK
      • Fink DS
      • Clary MS.
      The effect of vocal fold augmentation on cough symptoms in the presence of glottic insufficiency.
      Yang et al. (2021) is the only behavioral therapy study that included the CSI. They investigated the effects of two-to-four sessions of in-person breathing training plus cough suppression therapy delivered over six months. Patients in that pre-post study had an average decrease in the CSI of -6.7 from a baseline mean of 16.74.
      • Yang J
      • Crawley B
      • Dehom S
      • et al.
      Chronic refractory cough: objective improvement with aerodynamic-focused.
      Although the CSI decrease of -4.6 in the current study is slightly lower, the baseline mean CSI score in the present study was higher at 20.98, suggesting patients in the current study were more severe. Factors such as differences in the intervention and duration of treatment may also account for the slightly greater improvement in the Yang study. Of note, none of the studies that used the CSI as a cough outcome measure achieved a final CSI score of equal to or less than 3.23 (considered the normal cutoff for being cough symptomatic), indicating that despite showing improvement, on average, participants were still registering a symptomatic cough.
      While the study was not balanced to analyze the effects of sex on cough outcomes, it is worth noting that the six male participants did not demonstrate as robust a CSI decrease as the females. Closer investigation of these male participants revealed one, who had a negative response to treatment (Pre CSI: 19, Post CSI: 32), was referred to gastroenterology (GI) after reporting signs and symptoms consistent with reflux over the course of BCST. A confirmatory BRAVO was completed with results demonstrating a high correlation between cough and reflux events. Per recent interaction with GI, the patient's enhanced compliance with reflux therapy has since eliminated the cough. It should be noted that GI referral was generated by the treating SLP during BCST. This may indicate that BCST remains an appropriate intervention for cough that provides not only treatment but also ongoing evaluation. Three other male participants described a qualitative difference in their symptoms either during a follow-up physician visit or final BCST session with the SLP; these changes, however, were not represented by the CSI score. Further study with a larger sample of males is necessary to determine if there are sex-specific responses to BCST delivered via telehealth specifically.
      No such outliers were observed in the female group.
      Limitations. The current study has several limitations. One, the MCID for the CSI is unknown. Results of the current study indicate statistically significant improvements, but how clinically meaningful those improvements were is unknown. Future clinical practice and cough research would benefit from the use of an outcome measure in which the MCID is known (eg, the LCQ). Second, the sex differences observed in this investigation might be biased due to unbalanced sample size between the two groups. The study had significantly more females than males, so it is unknown if the small change in CSI in the males is representative of the greater population of males with RCC. The male group also had at least one participant whose cough resolved with GI evaluation and treatment, and several participants whose subjective response to treatment (verbal report of improved cough) did not align with their post-treatment CSI scores. Third, many patients who received evaluation and some treatment for RCC were not included in this retrospective review because they did not have complete patient-reported outcome information, either because it was not recorded, or they were lost to follow-up. At the time of data collection, patient-reported outcome measures were collected via an online portal system. Prior to each visit, patients received a text message from the healthcare system requesting they complete the forms. Incomplete data can result from patients opting out of text alerts or ignoring them. The absence of complete clinical data may contribute to poor understanding of outcomes for the wider patient population who either self-discharge or complete BCST without completing a post-treatment patient-reported quality-of-life measure (eg, CSI). Fourth, it is not possible to compare the findings of the current study to in-person data from the same location, because the treating location did not historically collect CSI data with regularity before introduction of an automated patient-reported outcome measures (PROMS) survey system, which was put into effect in the beginning of 2020 and no-longer provides in-person BCST secondary to concerns related to aerosolized respiratory particles. Fifth, while BCST was delivered by five highly skilled, specialized SLPs there was no standardized, prescriptive therapeutic approach to care, as seen in the Yang (2021) article. Finally, these data are from one clinic in an urban setting in the US Southeast. Future research should include more geographically diverse populations.

      CONCLUSION

      This study provides evidence that BCST delivered via telehealth improves cough-related quality-of-life in patients with RCC. Providers and patients should not avoid virtual delivery of BCST as it is low-cost, low-risk, convenient, and, as this study demonstrates, effective in treating RCC. Figure 1

      Financial Disclosure

      The authors have no financial interests to report.

      Conflict of Interest

      The authors have no conflicts of interest to declare.

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