The Intensive Dysphagia Rehabilitation Approach Applied to Patients With Neurogenic Dysphagia: A Case Series Design Study

Accepted Manuscript The Intensive Dysphagia Rehabilitation approach applied to patients with neurogenic dysphagia: a case series design study Georgia A. Malandraki, PhD, Akila Rajappa, MS, Cagla Kantarcigil, MS, Elise Wagner, MS, Chandra Ivey, MD, Kathleen Youse, PhD PII:

S0003-9993(15)01502-6

DOI:

10.1016/j.apmr.2015.11.019

Reference:

YAPMR 56395

To appear in:

ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION

Received Date: 18 August 2015 Revised Date:

23 November 2015

Accepted Date: 26 November 2015

Please cite this article as: Malandraki GA, Rajappa A, Kantarcigil C, Wagner E, Ivey C, Youse K, The Intensive Dysphagia Rehabilitation approach applied to patients with neurogenic dysphagia: a case series design study, ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION (2016), doi: 10.1016/j.apmr.2015.11.019. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

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Running head: The Intensive Dysphagia Rehabilitation

neurogenic dysphagia: a case series design study

Authors in order of Authorship

Akila Rajappa2, MS

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Georgia A. Malandraki1, 2, 3, PhD

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Title: The Intensive Dysphagia Rehabilitation approach applied to patients with

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Cagla Kantarcigil1, 2, MS Elise Wagner2, MS

Chandra Ivey4*, MD

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Kathleen Youse2, PhD

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Department of Speech, Language and Hearing Sciences, Purdue University

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Department of Biobehavioral Sciences, Teachers College, Columbia University,

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New York

Dysphagia Clinic, Hospital Evangelismos, National and Kapodistrian University

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of Athens, Greece

Department of Otolaryngology/Head and Neck Surgery, Columbia University

* Now at Department of Otolaryngology, Icahn School of Medicine, Mount Sinai

Presentation Disclosure: Part of these data has been presented at the Annual Convention of the American Speech Language and Hearing Association in 2014.

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Funding Source: This work has been partially supported by Teachers College, Columbia University and by a seed grant awarded to the first author by Purdue

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University.

Acknowledgements: The authors wish to thank Jaime Bauer Malandraki, MS,

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CCC-SLP for her assistance in data collection and analysis; and Professor

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Jessica Huber, Ph.D., CCC-SLP, for her valuable comments on the manuscript.

Financial Disclosure: The authors had been receiving salaries by their institutions during the study completion.

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Corresponding Author and Author for Reprint Requests: Georgia A. Malandraki, PhD, CCC-SLP, BCS-S

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Dept. of Speech, Language and Hearing Sciences Purdue University 715 Clinic Drive / Lyles-Porter Hall Room 3152 West Lafayette, IN 47907 Tel: 765-496-0206 E-mail: [email protected]

Conflicts of Interest: Dr. Malandraki developed IDR. None of the other authors has any conflict of interests.

Reprints are not available.

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Title: The Intensive Dysphagia Rehabilitation approach applied to patients with

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neurogenic dysphagia: a case series design study

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Abstract

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Objective: To examine the effects of the Intensive Dysphagia Rehabilitation

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(IDR) approach on physiological and functional swallowing outcomes in adults

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with neurogenic dysphagia.

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Design: Intervention study; before-after trial with 4-week follow-up through an

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online survey.

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Setting: Outpatient university clinics.

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Participants: A consecutive sample of 10 subjects recruited from outpatient

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university clinics. All were diagnosed with adult-onset neurologic injury or

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disease. Dysphagia diagnosis was confirmed through clinical and endoscopic

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swallowing evaluations. No subject withdrew from the study.

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Interventions: Participants completed the 4-week Intensive Dysphagia

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Rehabilitation, including: two oropharyngeal exercise regimens, a targeted

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swallowing routine using salient stimuli, and caregiver participation. Treatment

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included hourly sessions twice/week, and home practice for ~45 minutes/day.

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Main Outcome Measure(s): Outcome measures assessed pre- and post-IDR

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were: (1) airway safety using an 8-point Penetration Aspiration scale; (2) lingual

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isometric pressures; (3) self-reported swallowing-related quality of life (QOL), and

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(4) level of oral intake. Also, patients were monitored for adverse dysphagia-

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related effects. QOL and adverse effects were also assessed at the 4-week

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follow-up (online survey).

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Results: IDR was effective in improving maximum and mean Penetration

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Aspiration scale scores (p<0.05, η2=0.8146; p<0.05, η2=0.799708); and level of

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oral intake (p<0.005, Cohen’s d=-1.387). Of 5 patients who were feeding tube

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dependent initially, two progressed to total, and two to partial oral nutrition. One

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remained tube dependent. QOL was significantly improved at the 4-week follow-

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up (95% CI [6.38, 14.5], p<0.00), but not at the post-IDR assessment. No

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adverse effects were observed/reported.

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Conclusion(s): We conclude that IDR was safe and improved physiological and

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some functional swallowing outcomes in our sample, however further investigation is needed before it can be widely applied.

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Key words: deglutition, deglutition disorders, rehabilitation, neuroplasticity

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List of abbreviations (in alphabetical order):

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ASHA American Speech-Language-Hearing Association

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EAT-10 Eating Assessment Tool

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FEES Fiberoptic Endoscopic Evaluation of Swallowing

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IDR Intensive Dysphagia Rehabilitation

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MOST Multiphase Optimization Strategy

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NOMS National Outcomes Measuring System

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NPO nil per os (nothing by mouth)

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PAS Penetration Aspiration Scale

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PO per os (by mouth)

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PPO pars per os (partially by mouth)

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QOL quality of life

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MANUSCRIPT BODY

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Oropharyngeal dysphagia can compromise quality of life, nutrition and health; therefore, its timely and efficient management is crucial. Typical

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dysphagia management includes compensatory measures to protect the airway,

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such as postural adjustments, airway closure maneuvers, and diet modifications,

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which produce temporary effects and have poor patient adherence.1,2 Behavioral

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swallowing rehabilitative treatments such as strengthening or range-of-motion

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exercises3–11 are also frequently used and have been the focus of recent

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research, mainly because they address the underlying physiological deficits and

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thus can have a long-lasting effect.

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Rehabilitation of sensorimotor function, such as swallowing, after brain

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damage, can occur best through experiences that modify brain’s structure and

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function, a.k.a. experience-dependent brain plasticity.12,13 Existing rehabilitative

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swallowing treatments typically entail single exercise regimens involving the head

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and neck,3–11 and are based on few principles of experience-dependent plasticity

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and/or exercise physiology guidelines. Some of these treatments follow an

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intensive schedule and have showed feasibility and positive swallowing

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outcomes for neurogenic dysphagia.3,5,7,11 Furthermore, recent reimbursement

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reductions and Medicare therapy caps make it difficult to provide dysphagia

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treatment for long periods, thus encouraging research on intensive treatment

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approaches.

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Given the high complexity of swallowing, single exercise regimens, even when performed intensively, may be inadequate to rehabilitate the complex

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swallowing abnormalities typically seen in patients with moderate or severe

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dysphagia.14 Indeed, in a study presenting results from a web-based survey

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completed by >200 clinicians, it was found that clinicians use more than four

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swallowing exercises/interventions per session, in order to maximize patient

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outcomes in the short durations currently reimbursed by insurance.15 This is

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implemented, however, with great variability with respect to treatment

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combinations, frequency, and intensity, and little (if any) efficacy data are

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collected.15

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In response to the need to develop and examine intensive protocols that

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systematically combine interventions in an evidence-based manner, we

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developed a neuroplasticity-driven treatment approach, known as Intensive

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Dysphagia Rehabilitation (IDR). IDR incorporates a) evidence-based

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oropharyngeal training increasing gradually in intensity based on exercise

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physiology guidelines, b) targeted swallowing practice increasing gradually in

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complexity following principles of experience-dependent brain plasticity, and c)

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adherence-inducing features. In the present study, we present this new approach

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and the findings of a case series aiming to examine the safety and effects of IDR

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on physiological and functional swallowing outcomes in patients with neurogenic

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dysphagia.

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1 METHODS

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Participants

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Ten consecutive adult patients who were referred to two outpatient

University specialty clinics and who fulfilled the inclusion criteria were recruited

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over three years (2012-2015) and participated. All neurologic patients referred to

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the clinics were examined for eligibility (n=43). Inclusion criteria were: a) adult-

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onset neurogenic etiology of dysphagia (determined by a neurologist); b)

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diagnosis of oropharyngeal dysphagia as determined by one or more of the

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following: a score>3 on the Penetration Aspiration Scale (PAS),16 a validated 8-

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point scale documenting level of airway invasion by food/liquid; a score of≤4 on

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the American Speech-Language-Hearing Association (ASHA) National

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Outcomes Measuring System (NOMS),17 a 7-point ordinal scale documenting the

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level of oral intake; and/or other endoscopic evidence of dysphagia, such as

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post-swallow residue, poor secretion management, delayed swallow initiation, or

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reduced pharyngeal constriction; c) willingness and cognitive ability to participate

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in the protocol (cognitive ability was informally assessed by our team); d) medical

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stability; and e) caregiver’s willingness to participate. Exclusion criteria included:

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inability to provide consent; inability to elicit a swallow or open the upper

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esophageal sphincter; history of head/neck surgery or radiation; and/or currently

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undergoing swallowing or speech treatment. The Universities institutional review

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boards approved this study, and all subjects provided written consent.

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Outcome Variables (Evaluations) and Instrumentation

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Pre- and Post-Intervention Assessments and 4-week Follow-up:

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The following physiological and functional outcome variables were

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assessed pre- and post-treatment: (1) airway safety via the 8-point PAS16; (2)

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anterior and posterior lingual isometric pressures using the Iowa Oral

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Performance Instrument (IOPIa); (3) self-reported swallowing-related quality of

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life (QOL) using the Eating Assessment Tool (EAT-10)18; and (4) functional level

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of oral intake using the ASHA NOMS17. In addition, throughout the study safety

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was evaluated by monitoring patients for adverse dysphagia-related effects, such

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as respiratory infections, compromised health status, and pneumonia-associated

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hospitalizations. QOL and adverse effects were also assessed four weeks post-

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treatment via an online survey.

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Airway Invasion: Airway safety was the primary outcome and was rated

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using the 8-point PAS (1 indicating no penetration/aspiration and 2–8 indicating

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increasing degrees of penetration/aspiration)16 during Fiberoptic Endoscopic

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Evaluations of Swallowing (FEES).19 FEES assessments were video-recorded

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and de-identified for analysis. During FEES, two trials of four bolus types were

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administered: 5ml thin liquid; 10ml thin liquid; thin liquid via straw (self-

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administered) and 5cc of pudding. For one patient, only one bolus type was

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administered (i.e., 5ml thin liquid) due to gross aspiration noted on first swallows.

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Two blinded (to treatment and time of assessment) reviewers scored all

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swallows. Ten percent of the swallows were also rated by the PI (first author).

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Lingual Pressures: Lingual manometric pressures for the anterior and

posterior tongue were obtained via the IOPIa, a handheld device with an air-filled

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bulb. Patients were instructed to press the bulb between the tongue and the hard

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palate. Baseline maximum pressures (1 repetition maximum)20 were obtained for

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each tongue location (anterior/posterior) by obtaining two sets of data that differ

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by less than 5% to account for variability.8,11

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Quality of life was assessed with the EAT-10,18 a validated self-

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assessment examining the effects of dysphagia on QOL. It includes ten

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statements and individual responses are scored from a range of 0-4, 0 indicating

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“no problem” and 4 indicating a “severe problem.” Total scores range from 0-40;

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higher values indicate more impaired QOL.

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Swallowing Functional Outcome: Swallowing functional outcomes were

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rated using the ASHA NOMS.17 ASHA has developed a Functional

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Communication Measure for swallowing as part of the National Outcome

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Measurements System, used by the Centers for Medicare and Medicaid Services

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Physician Quality Reporting System.17 The 7-point NOMS scale for swallowing

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(ranging from Level 1=nothing by mouth, to Level 7=safe and efficient

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swallowing) is commonly used in healthcare to document outcomes. NOMS were

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rated by two certified SLPs (with>3 years experience with dysphagia care) who

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were not involved in IDR delivery, had access to all evaluation data, and were

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blinded to time of assessment.

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6 Intervention

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Treatment Sessions: Subjects participated in hour-long treatment sessions twice/week, and practiced their exercises at home daily for approximately

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45minutes. The decision to have two in-clinic sessions/week and the remainder

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treatment at home was based on logistical/feasibility reasons, and to ensure

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generalizability with typical outpatient dysphagia management where patients are

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seen in person once or twice/week. Exercises included: two oropharyngeal

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exercise regimens and one targeted swallowing practice (TSP) routine. Prior to

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treatment initiation, the PI reviewed the evaluation data and patients’ case history

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to determine the exercises and the TSP routine for each subject based on four

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criteria: their underlying swallowing pathophysiology; their current and prior

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respiratory and general health status; their ability to perform the selected

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exercises accurately, and their potential to consume the least restrictive TSP

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safely (as determined during FEES). All subjects received the same amount of

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therapeutic contact, and IDR components (see below) were applied uniformly.

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However, patients’ treatment plans were individualized to address their specific

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deficits.

3 Protocol Description

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IDR is an intensive four-week treatment approach that is based on three

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components, detailed below.

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High intensity structured oropharyngeal training: Two evidence-based exercise regimens were selected for each patient (Table 1). Only two regimens

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were used to increase patient adherence and accurate completion. Based on the

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exercise principle of increased load,20 intensity for each regimen was increased

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gradually in one of the following ways: by weekly or biweekly re-evaluating

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maximum strength and increasing training target goals; by weekly or biweekly

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increasing the number of repetitions of each exercise set (i.e., increasing volume

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of practice); or by increasing duration of each repetition (i.e., increasing volume

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of practice).11,20,21 In addition, for maximum benefit, each regimen targeted

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different muscle groups (e.g., lingual, pharyngeal, or suprahyoid) or had different

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neuromuscular goals (e.g., strength vs. range-of-motion). The selected regimens

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were practiced on alternating days to allow muscle rest/recovery,20,22 reduce

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fatigue, and maintain subject motivation.

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High intensity targeted swallowing practice (TSP): Patients also completed a daily TSP routine including single swallows of materials identified during FEES

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as least restrictive while still allowing for targeted/challenging practice. Patients

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were instructed to practice 60 swallows/day (in sets of 20, three times/day)

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unless they were deemed unsafe and were asked to start with 30 swallows/day.

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TSP was implemented to allow: continued use of the swallowing mechanism and

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the central and peripheral neural circuits engaged in swallowing (per the “use it

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or lose it” principle of experience-dependent plasticity12); and training specificity

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(per the specificity principle of experience-dependent plasticity and motor

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learning).23 Advancement or downgrading of materials during TSP was

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determined by patient performance. This refers to clinical observations relating

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to: duration of oral preparation time, approximate timing of the initiation of the

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swallow, potential expectoration of the material, overt clinical signs of aspiration,

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observed oral or oropharyngeal residue after each swallow, overall duration of

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each swallow, and respiratory function as monitored with pulse oximetry.

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Respiratory function was monitored via pulse oximetry during all in-clinic

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sessions for most patients and for some patients (for whom we had more

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concerns), respiratory function was also monitored at home. In general, if

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consistent difficulties (across multiple trials) were observed in any three of the

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following parameters, i.e., signs of aspiration, expectoration, and respiratory

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function, then we would consider downgrading materials. This was, however,

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very infrequent. In addition to this protocol, all patients were in stable health

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condition and were also instructed to follow rigorous oral care during and after

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IDR, because a main concern associated with aspiration is the aspiration of oral

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bacteria that may lead to pneumonia.24

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1 Adherence-inducing features: To encourage adherence, IDR included

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salience, social support, and short duration. Participants practiced swallowing

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salient foods, liquids or ice chips, i.e., flavors they found rewarding. Furthermore,

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caregivers participated in the in-clinic sessions and became patients’ “coaches”

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for home practice. Also, the protocol duration was short. In addition, caregivers

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and patients were provided with a binder including daily logs with step-by-step

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instructions and exercise log sheets. To encourage accurate completion of the

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home sessions, the following procedures were followed: a) the patients and the

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caregivers were instructed to complete the logs together (so both would be

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accountable), b) at the beginning of the in-clinic sessions, the clinician asked the

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patients and caregivers to review their log and the exercises/tasks performed

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during the previous day/s, and made corrections/adjustments to the logs, if

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needed, and c) at random times (once per week), the clinicians contacted the

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patients and caregivers (via phone or email) to ask questions and check whether

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the protocol was completed as prescribed for that particular day.

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Statistical Analysis

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Statistical tests were completed in SPSSb version 22. For the primary

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outcome variable (PAS scores), mean differences between pre-post scores were

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examined using the non-parametric Wilcoxon paired sign-rank test, because

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these values were available for 7/10 participants. Three patients declined

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participation in the post-treatment FEES, and were not included in the statistical

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analysis for this measure. For the EAT-10 scores, collected at three time points

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(pre-, post-intervention and at follow-up), means and standard deviations were

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calculated first. Then, a one-way repeated measures ANOVA was completed

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after data were tested for normality, using the Shapiro-Wilk’s test, and sphericity,

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using Mauchly’s Test of Sphericity and were found to be normally distributed

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(p>0.05) and to not violate the sphericity principle (x2(2)=2.398, p=0.302). Lastly,

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for the ASHA NOMS, pre- and post-treatment differences were examined using

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paired t-tests. Effect sizes were calculated using the η2 for the Wilcoxon and

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ANOVA tests and the Cohen’s d for the paired t-test comparisons.

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Insert Table 1 here

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RESULTS

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Demographics

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Demographics are presented in Table 2. Eight of ten patients were men

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(mean age:64.6±14.5). Six patients presented with a stroke, two had suffered a

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TBI and two had a peripheral nerve virus and a progressive neuromuscular

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disease, respectively (Table 2). Also, six patients presented with chronic

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dysphagia (dysphagia present post neurologic event/diagnosis that occurred >6

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months ago), and four with acute dysphagia (neurologic event/diagnosis

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occurred <6 months ago). All six patients with chronic dysphagia had received

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swallowing interventions in prior months or years with minor or no improvements.

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Five patients were NPO (nothing by mouth) and three patients were PPO

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(partially by mouth) at baseline.

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9 Patient Adherence

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Patient adherence was calculated as the percentages of completion for each exercise regimen and TSP, as it was documented in the everyday logs.

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Logs were evaluated for adherence weekly. The total average patient adherence

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percentages were 89.5%±10.04 (range:70-100%), suggesting overall good

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patient adherence.

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Penetration-Aspiration Scale Scores

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Figures 1 and 2 present the mean pre-post average and maximum PAS

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scores, respectively. Results showed a statistically significant decrease from pre-

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to post-treatment in the average PAS scores (z=-2.366, p<0.05, η2=0.799708),

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and the maximum PAS scores (z=-2.388, p<0.05, η2=0.8146) for the seven

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patients who participated in both pre and post-FEES, indicating improved airway

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safety.

3 Insert Figures 1 and 2 here

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Pre-post lingual isometric pressure changes are presented separately for

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patients who were engaged in lingual strengthening as part of IDR (N=4) (Table

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Lingual isometric pressures

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3,Part A) and for patients who were not enrolled in lingual strengthening (N=6)

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(Table 3,Part B). These results are presented descriptively. All but one patient

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increased in both anterior and posterior tongue pressures. Increases ranged from

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5 to 31kPa anteriorly and from 3 to 18kPa posteriorly for those who participated

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in lingual strengthening. Increases ranged from 4 to 9kPa anteriorly and from 2 to

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5kPa posteriorly for 5/6 remaining patients. One patient presented with minimally

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reduced (by 1kPa) pressures post-IDR.

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Insert Table 3 here

EAT-10

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Figure 3 presents the EAT-10 scores for each patient at three time points

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(pre-, post-treatment, and 4-week follow-up). One patient (Pt 7) did not respond

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to the 4-week follow-up survey. IDR elicited statistically significant changes on

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EAT-10 scores over time (F(2,16)=17.702, p<0.0005, η2=0.689)). Post hoc

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analysis with a Bonferroni adjustment revealed a statistically significant mean

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decrease of 10.44 points (95% CI [6.38, 14.5], p<0.00) from pre-treatment

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(mean=27.7, SD=5.99) to follow-up (mean=17.3, SD=8.0). Although mean EAT-

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10 scores also decreased from pre-treatment to post-treatment (mean=6.38,

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95%CI [-0.5, 12.5], p=0.072) and from post-treatment to follow-up (mean=4.44,

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95%CI [-0.6, 9.5], p=0.089), these differences were not statistically significant. In

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addition, despite the improvement in swallowing-related QOL across time, EAT-

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10 scores remained deviant from normal limits (i.e., >3/40).18

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Insert Figure 3 here

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ASHA NOMS and PO Status

A pre-post comparison on ASHA NOMS levels indicated significantly

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higher post-treatment levels (t(9) =-4.385, p<0.005, Cohen’s d=-1.387) (Figure

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4). Furthermore, of the five patients who were NPO at baseline, only one

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remained NPO (with pleasure trials orally) post-IDR. Two became fully PO (by

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mouth) (with some restrictions and strategies) and one started receiving most of

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his nutrition (>50%) by mouth. Of the three patients who were PPO, two

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remained PPO but were able to consume more foods and consistencies post-

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IDR, and one became fully PO.

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1 Insert Figure 4 here

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DISCUSSION

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The purpose of the present investigation was to examine the safety and

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preliminary effectiveness of a neuroplasticity-driven intensive treatment approach

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for neurogenic dysphagia. Our findings suggest that IDR was safe and improved

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airway safety and some functional swallowing outcomes in the patients

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examined. Swallowing-related QOL continued to improve four weeks after

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treatment. No dysphagia-related complications were observed/reported during

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treatment or at the follow-up.

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Specifically, improvements were documented via physiological (PAS

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scores and lingual pressures) and functional outcomes (EAT-10 and ASHA

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NOMS). Regarding PAS scores, of the seven patients who participated in both

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FEES, all improved in the PAS, indicating improved airway safety post-IDR.

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Improvements in airway safety have been documented in patients with dysphagia

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after single exercise regimens3,7,11; however, most of these regimens have had

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longer durations (six to eight weeks),7,11 strict participation eligibility criteria,7,11 or

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required daily in-clinic visits.3 IDR appeared to be effective in significantly

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improving airway safety in four weeks without the need of daily in-clinic therapy.

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This is likely due to the cumulative effect of IDR components, including the

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individualized and systematic combination of evidence-based exercises, the

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functional TSP enhancing training specificity, and the inclusion of adherence-

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inducing features absent from existing regimens.

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Furthermore, increased lingual pressures were noted post-IDR in 9/10

patients. For patients completing lingual strengthening (N=4), lingual pressure

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gains were pronounced (25%-225%) and are consistent with or higher than those

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reported previously in patients post-stroke.11 For patients who were not

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completing lingual strengthening, most were engaged in pharyngeal

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strengthening and/or hyolaryngeal elevation exercises. The small strength gains

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(8.5%-29%) in these patients may be explained by the shared musculature

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(lingual and suprahyoid) that is stimulated with these exercises and is known to

14

contribute to these pressures.4

16

Nine participants had improved EAT-10 scores post-IDR. However, these

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improvements were not statistically significant. At the 4-week follow-up patients

18

reported further improved QOL compared to baseline, which was significant. This

19

may suggest a carryover effect associated with generalization of behaviors

20

through daily use (i.e., swallowing). One patient had a higher/worse total EAT-10

21

score immediately post-IDR, but continued to report improved scores at follow-

22

up. This patient had baseline NPO status for three years prior to IDR, so it is

23

surmised that once he started receiving some oral nutrition, realization of what he

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was missing may have influenced his post-IDR QOL perception. It is also

2

important to note that although all patients had QOL improvements at follow-up,

3

none reported a score of <3, which would signify normal swallowing-related

4

QOL.18 This may indicate that IDR’s duration is insufficient to return patients with

5

severe dysphagia to fully normal QOL.

6

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Our most important findings relate to ASHA NOMS level reductions.

NOMS levels were improved for 8/10 participants and remained unchanged in

8

two. One participant who maintained his level had been diagnosed with a chronic

9

progressive condition, and improvements after any exercise would be unlikely.

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This patient also had the lowest adherence rates. The other patient was

11

diagnosed with an unspecified peripheral nervous system virus, and was three

12

months post-diagnosis. Remarkably, of the five patients who were NPO at

13

baseline, only one remained NPO (with pleasure trials orally) post-IDR. This

14

patient was diagnosed with a brainstem stroke (3 years post-onset) and was

15

unable to consistently trigger a swallow at baseline. Post-IDR, he gained the

16

ability to more consistently initiate a swallow and manage his secretions, and

17

started accepting some soft foods by mouth (pleasure feedings). Two of the five

18

became fully PO and one started receiving most of his nutrition (>50%) by mouth.

19

These findings indicate significantly improved functional swallowing outcomes

20

post-IDR.

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Study Limitations

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1

This study is a case series with a limited sample size. Case series designs can provide important information when testing new treatment models and

3

determining their safety and effectiveness; however, their results may not

4

generalize to the larger population. Also, at this time, compliance of the home

5

sessions was measured through patient/caregiver report (logs). In the future,

6

compliance and home sessions fidelity should be examined via home visits,

7

videotaped sessions or tele-sessions.

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2

Additionally, we acknowledge that recovery and responsiveness to

10

intensive therapy may be different across different diagnoses and across

11

different durations of neurological deficit. This is why IDR addresses each patient

12

with an individualized plan of care, although intensity and frequency remains the

13

same. At this exploratory stage, we included all qualifying patients in our

14

protocol, however, in the future each diagnostic category should be examined

15

separately. Furthermore, the personalized nature of IDR, with the inclusion of

16

different combination of exercises, complicates the interpretation of the findings.

17

The goal of this study was not to determine which exercise or combination was

18

more effective in improving swallowing outcomes. Our goal was to examine the

19

safety and preliminary effectiveness of a carefully designed individualized and

20

intensive treatment approach, representative of clinical practice patterns (where

21

many exercises are used), in optimizing treatment potential. Currently, we are

22

using the Multiphase Optimization Strategy (MOST)25 to identify the active

23

components of different IDR versions, in order to design randomized clinical trials

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1

for each version.

2 3

In addition, the use of FEES did not allow us to collect data on swallowing kinematics, such as hyolaryngeal excursion. Such measurements would

5

elucidate the underlying physiological parameters that were improved in our

6

patients and should be employed in future studies. Furthermore, the 4-week

7

follow-up included an online EAT-10 survey and online questions related to

8

adverse effects. Due to funding/staff restrictions, it was not possible to have

9

patients return for a thorough follow-up re-evaluation, but this should be

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addressed in the future. Lastly, despite the improvements seen in our patients,

11

some remained on restricted diets and all reported continued reduced QOL at

12

follow-up. This may indicate that the duration of treatment was inadequate.

13

Further research on optimal duration is warranted.

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CONCLUSION

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Our findings suggest that IDR improved physiological and some functional outcomes in the neurologic patients examined herein, but further evaluation is

19

needed before it can be generalized. These results form the basis for further

20

research investigations and future randomized clinical trials.

21 22

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Suppliers’ list:

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a. Iowa Oral Performance Instrument; IOPI Medical, LLC, 5901 Tolt River Rd

1

NE, Carnation, WA 98014.

2

b. SPSS Inc, 223 Wacker Drive, 11th Fl, Chicago, IL 60606-6412

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References:

1.

noncompliance with recommendations by a speech-language pathologist.

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Am. J. Speech-Language Pathol. 2005;14:61–70.

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Colodny N. Dysphagic independent feeders’ justifications for

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Macqueen CE, Taubert S, Cotter D, Stevens S, Frost GS. Which commercial thickening agent do patients prefer? Dysphagia. 2003;18:46–

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52. 3.

case-control study. Arch. Phys. Med. Rehabil. 2010;91:743–9. 4.

exercise conditions. Dysphagia. 2014;29:553–63.

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investigation of the mcneill dysphagia therapy program. Arch. Phys. Med.

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Rehabil. 2012;93:1173–8.

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Crary MA, Carnaby GD, Lagorio LA, Carvajal PJ. Functional and

physiological outcomes from an exercise-based dysphagia therapy: A pilot

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Clark H, Shelton N. Training effects of the effortful swallow under three

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Carnaby-Mann GD, Crary MA. McNeill Dysphagia Therapy Program: A

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Lazarus CL, Logemann JA, Huang CF, Rademaker AW. Effects of two types of tongue strengthening exercises in young normals. Folia Phoniatr.

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Logop. 2003;55:199–205.

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Logemann JA, Rademaker A, Pauloski BR, Kelly A, Stangl-Mcbreen C, Antinoja J, et al. A randomized study comparing the Shaker exercise with

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traditional therapy: A preliminary study. Dysphagia. 2009;24:403–11.

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Malandraki GA, Kaufman A, Hind J, Ennis S, Gangnon R, Waclawik A, et al. The effects of lingual intervention in a patient with inclusion body

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myositis and Sjogren’s syndrome: A longitudinal case study. Arch. Phys.

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Med. Rehabil. 2012;93:1469–75. 9.

McCullough GH, Kamarunas E, Mann GC, Schmidley JW, Robbins JA,

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Crary MA. Effects of Mendelsohn maneuver on measures of swallowing

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duration post stroke. Top. Stroke Rehabil. 2012;19:234–43.

12

10.

Robbins J, Gangnon RE, Theis SM, Kays SA, Hewitt AL, Hind JA. The effects of lingual exercise on swallowing in older adults. J. Am. Geriatr.

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Soc. 2005;53:1483–9.

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Robbins J, Kays SA, Gangnon RE, Hind JA, Hewitt AL, Gentry LR, et al. The effects of lingual exercise in stroke patients with dysphagia. Arch.

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Phys. Med. Rehabil. 2007;88:150–8.

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Kleim JA, Jones TA. Principles of experience-dependent neural plasticity:

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Implications for rehabilitation after brain damage. In: Journal of speech, language, and hearing research : JSLHR. 2008. p. S225–39.

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Robbins J, Butler SG, Daniels SK, Diez Gross R, Langmore S, Lazarus CL,

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et al. Swallowing and dysphagia rehabilitation: Translating principles of

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neural plasticity into clinically oriented evidence. In: Journal of speech,

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language, and hearing research. 2008. p. S276–300.

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acute poststroke patients. Semin. Speech Lang. 2013;34:154–69. 15.

A survey of usa dysphagia practice patterns. Dysphagia. 2013;28:567–74.

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Carnaby GD, Harenberg L. What is “usual care” in dysphagia rehabilitation:

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Rogus-Pulia N, Robbins J. Approaches to the rehabilitation of dysphagia in

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Rosenbek JC, Robbins JA, Roecker EB, Coyle JL, Wood JL. A penetration-aspiration scale. Dysphagia. 1996;11:93–8.

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Association AS-L-H. National Outcomes Measurement System (NOMS):

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Adult speech-language pathology user’s guide. Rockville, MD: ASHA;

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2003.

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Belafsky PC, Mouadeb DA, Rees CJ, Pryor JC, Postma GN, Allen J, et al. Validity and reliability of the Eating Assessment Tool (EAT-10). Ann. Otol.

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Rhinol. Laryngol. 2008;117:919–24.

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Butler S, Markley L, Sanders B, Stuart A. Reliability of the penetration aspiration scale with flexible endoscopic evaluation of swallowing. Ann Otol

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Rhinol Laryngol. 2015;124:480–3.

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et al. Quantity and quality of exercise for developing and maintaining cardiorespiratory, musculoskeletal, and neuromotor fitness in apparently healthy adults: Guidance for prescribing exercise. Med. Sci. Sports Exerc. 2011;43:1334–59.

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Garber CE, Blissmer B, Deschenes MR, Franklin BA, Lamonte MJ, Lee IM,

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Burkhead LM, Sapienza CM, Rosenbek JC. Strength-training exercise in

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dysphagia rehabilitation: Principles, procedures, and directions for future

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research. Dysphagia. 2007;22:251–65.

New Delhi; India: Jaypee Brothers; 1996.

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23.

Schmidt RA, Lee TD. Motor control and learning: A behavioral emphasis. 2005.

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Kisner C, Colby L. Therapeutic Exercise, Foundations and Techniques.

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Langmore SE, Terpenning MS, Schork A, Chen Y, Murray JT, Lopatin D, et

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al. Predictors of aspiration pneumonia: How important is dysphagia?

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Dysphagia. 1998;13:69–81.

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25.

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Collins LM, Murphy SA, Strecher V. The Multiphase Optimization Strategy (MOST) and the Sequential Multiple Assignment Randomized Trial

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(SMART). New methods for more potent eHealth interventions. Am. J.

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Prev. Med. 2007;32.

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Figure Legends:

17

Figure 1: Means and SDs of the Penetration-Aspiration Scale scores pre- and

18

post-IDR for each patient. * Pre-post group comparisons were significant at

19

p<0.05 (z=-2.366, η2=0.799708). Patients 1, 3 and 7 were not included in the

20

statistical analysis for this measure (as they did not have a post-IDR FEES).

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Figure 2: Maximum Penetration-Aspiration Scale scores pre- and post-IDR for

23

each patient. * Pre-post group comparisons were significant at p<0.05 (z=-2.388,

25

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1

η2=0.8146). Patients 1, 3 and 7 were not included in the statistical analysis for

2

this measure (as they did not have a post-IDR FEES).

3 Figure 3: EAT-10 scores pre- and post-IDR and at the 4-week follow-up (via

5

online survey) for each patient. * Pre-treatment to follow-up group comparisons

6

were significant at p<0.0005 (F(2,16)=17.702, η2=0.689).

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Figure 4: ASHA NOMS levels pre- and post-IDR for each patient. * Pre-post

9

group comparisons were significant at p<0.005 (t(9) =-4.385, Cohen’s d=-1.387).

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Table 1: Exercise regimens selected for each patient

Exercise Regimen A

Exercise Regimen B

1

Lingual Strengthening

Pharyngeal Strengthening

(use of IOPI*)

(effortful swallows with VAS†)

Lingual Strengthening

Pharyngeal Strengthening

(use of IOPI*)

(effortful swallows with VAS†)

Base of tongue (BOT)

Pharyngeal Strengthening

Range-of-Motion

(effortful swallows with VAS†)

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Patient

(tongue hold exercise with mirror)

Hyolaryngeal complex range-of-

(effortful swallows with VAS†)

motion and strengthening

Base of tongue (BOT)

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Pharyngeal Strengthening

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VAS†) Hyolaryngeal complex range-of-

Range-of-Motion

motion and strengthening

(tongue hold exercise with

(head lift exercise with timer)

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(Mendelshon maneuver with

mirror)

Lingual Strengthening

Pharyngeal Strengthening

(use of IOPI*)

(effortful swallows with VAS†)

Pharyngeal Strengthening

Hyolaryngeal complex range-of-

(effortful swallows with VAS†)

motion and strengthening

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(Mendelshon maneuver with VAS†) Pharyngeal Strengthening

Hyolaryngeal complex range-of-

(effortful swallows with VAS)

motion and strengthening

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8

(Mendelshon maneuver with VAS†)

Base of tongue (BOT)

Pharyngeal Strengthening

Range-of-Motion

(effortful swallows with VAS†)

mirror) 10

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(tongue hold exercise with

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Lingual Strengthening

Pharyngeal Strengthening

(use of IOPI)

(effortful swallows with VAS†)

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*IOPI: Iowa Oral Performance Instrument; † VAS: Visual Analog Scale ranging from 1 to 5 for effortful swallows [1=normal swallow/no effort, 5=the hardest possible swallow], and from 1 to 3 for the Mendelshon maneuver [1=1 second, 2=2 seconds, and 3=3 seconds holding Adam’s apple elevated]

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Table 2: Patient Demographics

Primary

Time

Diagnosis

post-

Age

Gender

of oral intake at

onset Brainstem

baseline

3 years

74

M

Multi-infarct

2 months

78

F

Stroke*

post last

2

NPO* (PEG†)

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NPO* (PEG†)

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Type of diet / level

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Patient

stroke Unspecified

4 months

cranial

post virus

nerve virus

infection

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3

62

F

Thin liquids and pureed diet

(left side)

1 year

Stroke*

post last

thick liquids as only

stroke

PO§ intake

MCA#

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5

PPO‡ with nectar

Multi-infarct

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4

5 months

86

63

M

M

Stroke*

PPO‡ with nectar thick liquids and pureed as only PO intake

6

Brainstem Stroke*

7 months

66

M

PPO‡ with pleasure PO§ of pureed food

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with small amounts of thin liquids 7 years

Dystrophy

post

and mechanical soft

diagnosis

diet

TBI||

4.5

47

36

months Brainstem

1.2 years

Stroke* 10

TBI||

M

Nectar-thick liquids

NPO* (PEG†) except for therapeutic trials

64

M

NPO* (PEG†) except for therapeutic trials

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Muscular

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8 months

70

M

NPO* (PEG†)

* NPO: nil per os (nothing by mouth); † PEG: Percutaneous Gastrostomy tube feeding; ‡ PPO: pars per os (partially by mouth); § PO: per os (by mouth); || TBI:

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Traumatic Brain Injury; # MCA: Middle Cerebral Artery

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Table 3: Maximum lingual pressures before and after IDR

Part A: Maximum Lingual Pressures Pre and Post Treatment

Post-IDR

Anterior

Anterior

Change

% of

in kPa

Change

Patient

20

25

5

25

Pt 2

11

26

15

136

Pt 6

16

47

31

Pt 10

22

34

12

Post-IDR

Posterior

Posterior

Change

% of

in kPa

Change

2

5

3

150

8

26

18

225

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Pt 1

Pre-IDR

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Pre-IDR

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(lingual strengthening part of treatment; N=4)

194

22

35

13

59

55

21

32

11

52

Part B: Maximum Lingual Pressures Pre and Post Treatment

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(lingual strengthening NOT part of treatment; N=6)

39

5

14

35

38

3

8.5

17

22

5

29

14

16

2

14

Pt 5

30

34

4

13

28

31

3

10.7

Pt 7

17

16

-1

-5.8

13

12

-1

-7

Pt 8

37

46

9

24

36

41

5

13

Pt 9

24

28

4

16

22

26

4

18

34

Pt 4

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Pt 3

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