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Clinical Case Studies 2020, Vol. 19(2) 101 –114

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The Successful Treatment of Insomnia in a Patient With a Complex Neurological History

James P. Loveless1 , Jaclyn M. Russo2, and Virginia C. Andersen2

Abstract Insomnia is one of the more common sleep–wake disorders from which people suffer. This is particularly true among individuals who have experienced neurological insult via conditions such as multiple sclerosis, stroke, and other neurodegenerative disorders. While cognitive- behavioral therapy for insomnia (CBT-I) is one of the most effective behavioral interventions for insomnia, there is a dearth of empirical literature on its application to patients who have a history of neurological disorders. The present case study illustrates a largely successful course of CBT-I to treat a persistent and severe case of insomnia for Eric, a 55-year-old Caucasian man with multiple sclerosis and a self-reported history of restless leg syndrome and stroke. His treatment course is described in detail, and the implications of this approach to care are discussed.

Keywords insomnia, CBT-I, RLS, multiple sclerosis

1 Theoretical and Research Basis for Treatment

Insomnia is the most common sleep disorder, with approximately one third of adults reporting dissatisfaction with sleep quality or quantity and difficulty initiating and maintaining sleep (American Psychiatric Association [APA], 2013; Grewal & Doghramji, 2016). Individuals with insomnia experience significant impairments in daytime functioning as evidenced by fatigue, sleepiness, impaired attention or concentration, mood disturbance, and impairments in occupa- tional, academic, interpersonal, and social functioning (APA, 2013). For these individuals, sleep difficulties occur at least three nights a week over a period of at least 3 months. Importantly, these challenges occur despite having adequate opportunity for sleep (APA, 2013). There are several risk factors for insomnia, including increasing age, gender (female), and physical and mental health difficulties (Grewal & Doghramji, 2016).

The research literature has yielded objective guidelines for what constitutes good sleep qual- ity. Optimal sleep duration for the average adult is approximately 6 to 8 hr per night (Youngstedt et al., 2017), with an average sleep latency, or the time it takes to fall asleep, being ≤30 min

1Middle Tennessee State University, Murfreesboro, TN, USA 2University of Virginia, Charlottesville, VA, USA

Corresponding Author: James P. Loveless, Middle Tennessee State University, P.O. Box 87, Murfreesboro, TN 37132, USA. Email: [email protected]

890123CCSXXX10.1177/1534650119890123Clinical Case StudiesLoveless et al. research-article2019

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(Ohayon et al., 2017). Average length of wakefulness periods after sleep onset (WASO) for those experiencing good sleep quality should be ≤20 min. Finally, a good sleep efficiency for adults, which is a measure that captures the percentage of time one spends in bed asleep, is defined as ≥75% (Ohayon et al., 2017). Individuals with insomnia often have low sleep efficiencies cou- pled with long sleep latencies and/or long WASOs. In addition, they can also experience other sleep maintenance difficulties such as persistent early morning awakenings where they wake up long before their desired wakeup time and are unable to fall back asleep.

Sleep-related problems have been found to be especially prevalent in individuals with neuro- logic conditions. In one study, individuals with multiple sclerosis (MS) reported significantly worse perceived sleep quality and were found to be overall poorer sleepers when compared with control participants without MS (Lunde et al., 2012). Furthermore, sleep disturbance among individuals with MS has been found to be associated with diminished visual, verbal, and working memory, executive function, attention, and processing speed (Braley & Boudreau, 2016). In another study with stoke survivors, more than 50% of participants developed insomnia within 4 months of experiencing the stroke (Dyken et al., 2012). In addition, individuals with neurologic conditions are at an increased risk of experiencing obstructive sleep apnea (OSA; Salam et al., 2019). Pillai and Leverenz (2017) propose several pathways linking neurodegenerative disease (NDD) neuropathology or neurologic conditions and sleep disturbance. Specifically, in this model, NDD neuropathology is proposed to disrupt individuals’ circadian network, sleep neural network, orexin/hypocretin system, and glymphatic clearance mechanisms (Pillai & Leverenz, 2017). Disruptions to these systems then impact individuals’ sleep/wake cycle, which in turn exacerbates sleep difficulties and perceived quality of sleep, thereby perpetuating the cycle of sleep disturbance. Effective treatment aims to intervene across these disrupted systems in indi- viduals with insomnia.

Cognitive-behavioral therapy for insomnia (CBT-I) is the gold standard in evidence-based treatment for individuals with insomnia. In a randomized clinical trial examining the dose– response effects of CBT-I, more than 50% of participants showed improvements at the end of the treatment, with the most optimal dose being four biweekly sessions (Edinger et al., 2007; Van Straten et al., 2018). CBT-I has also been shown to be effective for patients with both comorbid psychiatric and medical difficulties across diverse demographic groups (i.e., income, race, sex, age, education; Cheng et al., 2019; Siebern & Manber, 2011; Wu et al., 2015). Traditional CBT-I consists of five main components: (a) sleep restriction, (b) stimulus control, (c) cognitive therapy, (d) relaxation therapy, and (e) sleep hygiene (Winkelman, 2015). Sleep restriction aims to reduce time in bed to perceived total sleep time with the goal of increasing an individual’s sleep drive and stabilizing circadian rhythm. Stimulus control encourages individuals to only attempt to sleep when sleepy, to get out of bed after awakening in the night, and to use the bed only for sleep or sexual activity. The goal of stimulus control is to promote the association of bed and sleep. The cognitive component of CBT-I aims to restructure maladaptive beliefs regarding daytime and health consequences of insomnia by helping individuals to maintain reasonable expectations about sleep. Relaxation therapy helps to reduce physical and psychological arousal in the sleep environment through progressive muscle relaxation, breathing exercises, and/or meditation. Finally, CBT-I helps individuals to encourage positive sleep hygiene, which includes reducing behaviors that interfere with sleep drive or increase arousal (e.g., caffeine and alcohol intake, avoiding daytime napping). While some case examples exist in the literature (Clancy et al., 2015; Majendie et al., 2017), the effectiveness of CBT-I remains understudied among individuals with NDD, despite the prevalence of sleep disturbance among this population and the empirically based effectiveness of this treatment across diverse groups of individuals. Importantly, modifica- tions may need to be made to this protocol to best meet the needs of this population; however, the dearth of available research in this specific population presents a significant gap in the current literature. The case presented here offers some practical guidance for practitioners regarding

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modifications made to the CBT-I protocol for an individual with complex NDD and makes a call to the field for more rigorous research in this specific area of behavioral health interventions.

2 Case Introduction

The following case seeks to illustrate a course of CBT-I for a patient with a complex neurological history. Specifically, CBT-I was used to treat “Eric,” a 55-year-old Caucasian man with MS and a self-reported history of stroke who struggled with long-standing issues related to poor sleep initiation and maintenance. He was treated by a postdoctoral fellow and a practicum student co- therapist (who assisted in the intake, performed check-ins at the beginning of each appointment, and aided in the facilitation of problem-solving discussions as needed) in the context of a univer- sity health system behavioral medicine clinic. All work was supervised by an attending licensed clinical psychologist.

3 Presenting Complaints

Eric reported having a lifelong history of sleep disturbances that have persisted regardless of changes in mood or life circumstances. Specifically, he endorsed difficulties with both sleep initiation and maintenance. At the time of his intake appointment, he noted that he slept approxi- mately 2 to 3 hr, nightly, and that his average sleep latency was an estimated 1 to 2 hr. He was unable to provide an estimate of his average WASO; however, he remarked that his time awake at night “[felt] like forever.” He was not prescribed medication to help him with sleep at the time, though he reported that he had been previously prescribed Ambien and that it was somewhat helpful. His medical record indicated that he had also previously used melatonin to no noticeable effect. When asked about factors that could have been affecting his sleep, he identified chronic pain (related to MS and degenerative joint disease [DJD]) and nighttime anxiety (multiple anx- ious awakenings/night related to distressing dreams, some of which were reportedly related to physical/sexual/emotional abuse experienced as a child via his father). He also reported having a diagnosis of OSA and attempted to use his continuous positive airway pressure (CPAP) device regularly; though, he stated that he found the mask uncomfortable and had not yet noticed an improvement in his sleep quality while using it.

With regard to sleep hygiene, he stated that the bedroom environment was warm (no central AC; small window AC unit in living room), noisy (dogs barking outside), illuminated via a small nightlight, and contained multiple nonsleep-related distracters (a stereo and large television). He stated that he did not have a consistent routine to prepare for bed. He reported that he typically took his evening medications between 7:00 and 8:00 p.m. and then attempted to go to sleep between 8:00 and 8:30 p.m. His typical final wake time was between 7:00 and 8:00 a.m. He indicated that when he was unable to sleep, he would often stay in bed, and will sometimes watch TV. He denied purposefully engaging daytime napping but admitted that he would sometimes fall asleep in his power wheelchair due to its reclining ability. No maladaptive cognitions related to sleep were appreciated. A history of alcohol, tobacco, or other drug use was denied.

4 History

Medical History

Per Eric’s medical record, he carried diagnoses of MS, DJD, restless leg syndrome (RLS), sei- zure, mild OSA, Asthma, hypertension, hyperlidemia, and type 2 diabetes mellitus. He also reported a history of stroke (2×). With regard to his neurological history, most of his previous care was not completed at the current medical center, and much of what was known was based in

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his self-report. He stated that he was diagnosed with RLS and relapse-remitting MS in the early 1990s. At some point thereafter, he reportedly experienced two strokes, though his neurologist at the time of care in our clinic noted that there was inconsistent evidence to support that these occurred. Adherence to his medication regimen and CPAP therapy was described as “good,” and he reported having excellent attendance to his medical appointments; his medical record corrobo- rated his self-report. Eric ambulated with the assistance of a power wheelchair, as he reportedly lost the ability to walk during an MS relapse episode in the early 2000s. He had an in-home aid to help him with activities of daily living and more complex tasks around his apartment. Previous surgeries included Nissen fundoplication, eye surgery, and abdomen surgery. His family history was significant for coronary artery disease.

Psychiatric History

Eric reported that he has a long history of emotional lability with some sustained periods of “depression and anxiety.” Previous mental health diagnoses have reportedly included bipolar 1 disorder, anorexia nervosa, bulimia nervosa, as well as others that he was unable to recall. He stated that he has not struggled with eating pathology since adolescence. His mood at the time of his intake appointment was described as “fair.” He endorsed experiencing fatigue, avolition, and anhedonia. He denied the experience of daytime anxiety or re-experiencing symptoms related to his abuse. A history of manic symptomatology was denied. When asked about a history of psy- chotic symptomatology, he endorsed seeing the ghost of one of his deceased lovers in his apart- ment approximately 1 year ago. He was adamant that this was not a hallucination because his landlord reportedly witnessed it as well. A history of other aberrant visual phenomena, auditory hallucinations, and/or delusional thinking was denied. He also denied having suicidal ideation (SI) or homicidal ideation (HI). A history of HI was denied; however, he endorsed a history of SI with multiple suicide attempts (via overdosing and cutting) and hospitalizations for such. He was unable to remember the date of his most recent hospitalization, though he stated that he has not had an episode of SI in over a year. Eric also admitted to a history of nonsuicidal self-injury via cutting to cope with difficult emotions. His last use of such was reportedly 1 year ago.

At the time of the intake appointment, Eric was being followed by a psychiatrist in private practice and was prescribed Zyprexa (10 mg, qpm). He is also prescribed Depakote, Lyrica, and Baclofen by his other medical providers to manage his MS and seizure activity. Previous psychi- atric medications courses included Seroquel, Saphris, Lithium, and Ambien. He reported that involvement with the psychiatrist has been helpful. He was also being followed weekly/biweekly by a master’s level social worker for individual counseling to address ongoing stressors. He reported that this has also been helpful. He remained in care with both of these providers through- out his course of treatment in our clinic.

Family and Social History

Eric stated that he was from a mid-Atlantic state in the United States, and his family of origin includes his mother, father, brother, four sisters, and half brother. He described his childhood as abusive, noting that he suffered with physical, sexual, and emotional abuse perpetrated by his father from infancy to age 8. After turning 8 years old, his grandmother learned of the abuse and removed him from the family home at gunpoint. He lived with her until her death when he was 18 years old. He stated that his parents are still living. Notably, he reported having a continued relationship with his father despite “not trusting him.” When asked about his relationships with siblings, he stated that he was only close with his half brother. With regard to romantic relation- ships, Eric reported that he was a widower. He stated that he was married to his wife from 1997 to 2007, when she passed away. It is unknown how she died. He also reported having other

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deceased girlfriends, but these relationships were not discussed. He indicated that he did not have children.

Eric stated that he lived alone in a town approximately 1.5 to 2 hr travel time from the medical center. He endorsed having a few good friends whom he spent time with occasionally. Identified hobbies and interests include writing music, playing guitar, arts and crafts, jigsaw puzzles, and watching movies. He identified himself as a Christian and noted that his religious faith was very important to him.

Education and Employment Status

Eric stated that he had two bachelor’s degrees (one in computer science and the other in electrical engineering) and that he was working on an online master’s degree in music. He reported having a 30-year history of electrician work before becoming disabled approximately 14 years ago.

5 Assessment

The entire assessment process that Eric completed from referral to the start of intervention included an initial screening for active psychosis, suicidality, and homicidality by the clinic’s support staff, a brief review of the referral by the clinic director for appropriateness of fit for the clinic, an extensive medical record review by the receiving clinicians, an intake appointment that included a detailed semistructured interview, and baseline sleep data collection via sleep diaries. Normally, sleep-dis- order patients within this clinic do complete standardized measures related to sleep as a part of their intake appointment; however, Eric declined to complete pretreatment measures of insomnia sever- ity during his intake appointment due to not having his reading glasses, and the measures were not administered orally during the appointment because of time constraints within the clinic.

Ultimately, these objective measures of subjective sleep quality were not administered there- after because they were deemed unnecessary. At intake, he endorsed having a very poor subjec- tive sleep quality with daytime fatigue, long sleep latencies, and regular wakefulness after sleep onset. This self-report was corroborated by information in the medical record as well as by the objective data he produced when he completed his baseline sleep diary recordings. Additional indicators of poor subjective sleep quality would not likely have further informed our approach to care as they would have provided redundant information, so we elected to forego them. The rationale provided here is not meant to dismiss the role of standardized measures during the assessment phase of care, especially in cases where there is considerable degree of ambiguity created by conflicting data. As will be discussed later, the ambiguity Eric’s case rested in the complex etiology of his insomnia, rather than its relative presence or absence. Indeed, it was quite clear from the medical record, his self-report, and his sleep diary data that his insomnia was chronic and fairly severe.

With regard to objective sleep data, baseline data were gathered via sleep diaries over a 2-week period toward the beginning of care (see Table A1 of the appendix). Again, calculated measure of objective sleep quality was fairly consistent with his self-report at the time of his intake appointment with long sleep latencies and periods of wakefulness after sleep onset. Average total sleep times were less than 3 hr per night despite being in bed on average 9 to 9.5 hr per night. Sleep efficiency (which is computed by dividing average time in bed by average total sleep time) was very low.

6 Case Conceptualization

Consistent with Morin’s Three-Ps model of insomnia (Morin, 1993), the etiology and mainte- nance of Eric’s insomnia were thought to be multifactorial involving predisposing risk factors that were activated at one time by a provoking event or series of provoking events and ultimately

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maintained by a number of biological, behavioral, and environmental promoting factors that interfere with sleep. Given the reported lifelong duration of his insomnia and sheer number of factors involved, it was difficult to hypothesize about its exact etiology and course. Predisposing risk factors likely included genetic and other biological mechanisms tied to his abusive develop- mental history that primed him to have higher levels of baseline physiological arousal above and beyond what might be found among those from more normative backgrounds. Likewise, they may also include long-standing pathophysiological processes tied to his MS and RLS. His expe- riences with childhood physical and sexual abuse likely served as the initial provoking events for his insomnia, though it is very likely that other events such as episodes of MS disease progression and worsening of his RLS exacerbated his sleep difficulties. Finally, promoting factors that seemed likely to have maintained his insomnia included his history of poor sleep hygiene, long- standing patterns of behavior that have associated his bedroom environment with nonsleep- related activities, a bedroom environment not completely conducive to sleep, chronic pain, anxious arousal at night, neurological changes associated with his MS progression, and his use of Depakote, which has been shown to be disruptive to sleep initiation via increasing the amount of time patients spend in the first stage of sleep (Bazil, 2003; Zhang et al., 2014).

Overview of Goals and Treatment Plan

Eric identified that he wanted to improve his subjective sleep quality by increasing his overall sleep duration. CBT-I was selected because of its general effectiveness as well as its ability to target many of the behavioral and environmental factors that were likely impacting Eric’s sleep. Given his distance from the clinic, it was agreed that sessions would be scheduled on an approxi- mately biweekly basis. Including the intake appointment, he attended 13 sessions. Treatment progress was tracked via subjective report, sleep diary data, and posttreatment administration of sleep quality and daytime sleepiness measures.

7 Course of Treatment and Assessment of Progress

Psychoeducation and Introduction to Self-Monitoring

The first follow-up session following the intake appointment focused on providing sleep education and an overview of CBT-I. It also introduced Eric to self-monitoring via the sleep diary. Sleep edu- cation involved reviewing in detail handouts on sleep architecture, the Three-Ps model of insomnia, and circadian signaling. This information was then discussed in the context of Eric’s case, and the specific components of CBT-I were reviewed in detail. Research evidence on the effectiveness of CBT-I was discussed with an emphasis on which components tend to be associated with the greatest effect. After some further discussion, it was agreed that treatment would focus mainly on the behav- ioral components of the therapy with an as-needed focus on cognition. The session then shifted to an introduction to the sleep diary. He was oriented to how the data derived from it would be used to inform care, and then was taught how it was to be completed. He was instructed to complete it for homework, and for the sake of gathering good baseline sleep data, he was also instructed not to alter any of his normal sleep behaviors. The session was concluded with a review of the covered mate- rial. The teach-back method (wherein a patient relays back the information provided to them by the provider) was particularly useful in assessing for his level of understanding.

Relaxation Training and Creating a Bedtime Routine

The next session focused on providing relaxation training and planning for a soothing bedtime routine. Eric’s baseline sleep diary data were reviewed in detail with weekly averages being

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calculated. After discussion of those, the session transitioned to discussing his bedtime routine. His typical routine was very basic; he would take his evening medications between 7:00 and 8:00 p.m., transition from his wheelchair to his bed with the help of his in-home health worker, put on his CPAP device, and then attempt to go to sleep. The importance of a consistent and soothing 30- to 60-min routine prior to bed to prepare for sleep was discussed. Activities that could be included in this routine were brainstormed, and he identified prayer and reading his Bible as two activities that he could include. He was then introduced to diaphragmatic breathing as a strategy that could be incorporated into his routine. This was practiced in session to positive effect. The ordering of the behaviors in his bedtime routine was discussed, and he agreed to implement this new routine for homework in addition to keeping his sleep diary.

Stimulus Control and Sleep Hygiene Training

This session focused on improving sleep hygiene and stimulus control practices. Sleep diary data from the previous 3 weeks were reviewed. Overall reductions in both sleep latency and wakefulness after sleep onset were noted. Likewise, there was an increase in average total sleep time and sleep efficiency. These improvements were complicated by pain and narcotic use. Eric explained that shortly after the previous session, he broke two ribs after falling in his home while attempting to transfer himself from his wheelchair to his toilet. He attributed these improvements to the sedating effects of the brief course of pain medication that he was pre- scribed following the fall. He did endorse following through with the bedtime routine crafted during the previous session; however, he indicated that he was unable to use diaphragmatic breathing effectively due to the pain associated with his broken ribs. Progressive muscle relax- ation and guided imagery were introduced as alternatives to diaphragmatic breathing, and each was practiced within session. Eric ultimately selected guided imagery to use as an alternative to diaphragmatic breathing out of concern that progressive muscle relaxation might exacerbate muscle spasticity associated with MS.

After some further discussion of his bedtime routine, the session then shifted to exploring sleep hygiene and effective stimulus control strategies for sleep. Handouts were provided and reviewed in detail. Eric was able to identify several environmental variables that were not con- ducive to him getting good sleep. These included having an apartment next to a busy train track, noisy neighborhood animals, attempting to sleep with a nightlight, and having a poorly con- trolled bedroom temperature during the winter and summer months. In addition, he was able to identify behavioral patterns that were also likely disruptive to sleep including spending excessive amounts of time in bed when awake, engaging in nonsleep-related activities in the bedroom (i.e., watching TV, reading, and listening to music), and having inconsistent wake times.

Given the number of factors, several of these environmental and behavioral sleep disruptors were changeable while others would prove difficult to change. He was agreeable to wearing ear- plugs to reduce the effect of outside noises on his sleep, as well as to cease middle-of-the-night TV watching and music listening. Likewise, he was amenable to improving bedtime tempera- tures during more extreme months by utilizing fans during warmer nights and blankets as well as a space heater during colder nights. He noted that his finances necessitated continued living in an apartment next to the busy train track, and declined discontinuing the use of a nightlight.

With regard to his excessive time spent in bed, this was by necessity as he required the assis- tance of his in-home health worker to transition out of bed. Furthermore, she was only available to him between 7:00 a.m. and 8:00 p.m. each day. Stimulus control guidelines would have a patient avoid the use of the bed/bedroom for all activities not related to sleep, sex, and recovering from illness as a means of conditioning them to only associate the bed/bedroom with those activi- ties. Under regular circumstances, this would require a patient to cease all non-bedroom-related activities in the bedroom and perform them elsewhere. This also would extend to prolong periods

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of wakefulness. For Eric, this was not reasonable, so this guideline had to be modified to meet his needs. We worked to brainstorm an alternative set of environmental conditions that could dis- criminately be associated with sleep and wakefulness without him needing to leave his bed. Eric possessed an adjustable bed, and normally tried to sleep in a reclined position with the back of the bed being elevated at a 20° to 25° angle. We ultimately agreed that during periods of wakeful- ness, the back of his bed would be elevated at a steeper angle (~50°–60°), and that he would use a side-table lamp to increase the illumination in the room. We also brainstormed nonarousing activities that he could do during long periods of wakefulness in the evening. He identified read- ing his Bible and creating Christmas ornaments for loved ones as two nonarousing activities that he would likely do.

This session was concluded with a review of provided psychoeducation and the sleep hygiene/ stimulus control changes negotiated during the session. Eric indicated an understanding of what was discussed and expressed a willingness to attempt the discussed changes in addition to main- taining his bedtime routine as well as his self-monitoring.

Sleep Restriction and Subsequent Titration Sessions

This next session focused on beginning the process of sleep restriction. The session began with a review of his sleep diary, and the data revealed decreases in sleep latency and early morning awakenings with increases in total sleep time and sleep efficiency. He denied difficulties imple- menting sleep hygiene/stimulus control changes discussed during the previous session, and he indicated that he maintained the use of his bedtime routine. Psychoeducation on sleep restriction was then provided. The establishment of a consistent wake time was discussed (he selected 7:00 a.m. because it was the arrival time of his in-home health worker), and his earliest bedtime was negotiated based on sleep diary data from the previous week (12:30 a.m. was agreed upon). He agreed to maintain this earliest bedtime and latest wake time until his next appointment, as well as to continue the previously established behavioral changes around sleep.

The remaining seven sessions were spent titrating available sleep time in an effort to increase total sleep time and reduce sleep latency and wakefulness after sleep onset. Available sleep time was initially titrated down due to a spike in sleep latency and wakefulness after sleep onset, but these eventually decreased, and available sleep time was titrated up throughout the remainder of care. Sleep latency and wakefulness after sleep onset both plateaued at 60 min, and subsequent increases in total sleep time were due to increases in available sleep time. Titration of available sleep time ended after an improvement in self-reported subjective sleep quality. Throughout the sleep titration sessions, Eric maintained all of the previously discussed behavior changes. Maladaptive sleep-related cognitions were probed for, but he consistently denied having such.

Summary of Treatment Gains

Eric’s sleep had greatly improved over the course of care. Objectively, he had increased his overall sleep efficiency and total sleep time while decreasing his sleep latency, wakefulness after sleep onset, and early morning awakenings. His self-reported subjective sleep quality had also improved. He was administered the Insomnia Severity Index (ISI; Bastiem et al., 2001; Wong et al., 2017) and the Epworth Sleepiness Scale (ESS; Johns, 1991; Lapin et al., 2018) during his final session. The ISI is a standardized self-report instrument that measures insomnia severity across several domains, including difficulties with sleep initiation, difficulties with sleep maintenance, problems with early morning awakenings, sleep satisfaction, impact of the sleep problem on quality of life, distress caused by the sleep problem, and interference caused by the sleep problem in daily life. Scores can range from 0 to 28, with scores between 0 and 7 indicating no clinically significant insomnia (Bastiem et al., 2001). His score of 6/28 on the ISI indicated that he had experienced a

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minimal level of insomnia symptomatology within the 2 weeks prior to the session. Likewise, the ESS is a standardized self-report instrument that measures daytime sleepiness across a variety of everyday settings and scenarios. Scores can range from 0 to 24, with a score falling between 0 and 10 being considered indicative of a normal level of daytime sleepiness (Johns & Hocking, 2004). His score of 2/24 on the ESS indicated that he experienced a low level of daytime sleepiness at the time of the final session. A summary of his pre–post sleep diary data can be seen in Table A2 of the appendix. In addition, please refer to Figures A1, A2, and A3 of the appendix for graphs of his sleep diary data across his entire course. These tables and graphs can be found in the appendix.

8 Complicating Factors

Patient and Environmental Factors

There were several factors related to Eric’s complex medical and psychiatric histories as well as his environment that likely complicated care. For example, the motor deficits associated with his MS prevented him from being able to use many of the sleep hygiene and stimulus control strate- gies in a conventional fashion, and required a degree of creative problem-solving to overcome. Fortunately, he possessed resources (e.g., his adjustable hospital bed) that facilitated this process and seemed to help produce successful result. In addition, chronic pain and movement urgency associated with his MS, DJD, and RLS likely were disrupting influences for both sleep initiation and sleep maintenance throughout treatment. Likewise, nighttime anxious arousal associated with previous trauma exposure also likely interfered. His regular use of learned relaxation tech- niques, good adherence to his medication regimen, and engagement in his mental health care probably helped with these disrupting influences, but assuredly did not eliminate them com- pletely. The alterations to his sleep architecture likely caused by his adherence to Depakote was present throughout treatment, and may help to explain in part why greater gains were not observed in the area of sleep latency. Finally, there were factors in Eric’s home environment that posed challenges to his success in this treatment (e.g., difficulties with temperature management and his apartment’s proximity to an active railroad track). Issues related to temperature were easily addressed with fans, blankets, and heaters; however, the location of his home was unchangeable due to financial constraints. Train track activity was likely a major disruptor of sleep maintenance and will continue to be such until he relocates to an apartment or home away from a train track.

Therapeutic Relationship Factors

For the most part, Eric was an easy patient with which to work. During his intake appointment, however, there were concerns about boundary crossing as he commented on one of the therapists’ appearance by noting that they were “pretty.” He was easily redirected and after it was explained that this type of behavior was inappropriate, he did not make similar comments for the remainder of his care. Due to the lack of persistence of this behavior, it was thought of as perhaps a function of loneliness. Dovetailing with this hypothesis, he also seemed eager to please; homework was always completed and he was slow to admit to difficulties implementing such. Extra care and attention was paid to assessing for difficulties with understanding the presented material as well as problems following through with assigned homework.

9 Access and Barriers to Care

There were no major barriers that prevented Eric’s access to care. Due to his disability status, he possessed publicly subsidized health insurance and transportation to appointments through Medicaid. The distance of his home to the clinic as well as the sheer volume of his other medical

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appointments did limit the frequency that he was willing to attend appointments; nevertheless, he reliably attended his follow-up appointments on an approximately biweekly basis.

10 Follow-Up

No formal follow-up assessment was performed; however, at his final session, it was established that Eric could return for further care as needed to address any reoccurrence of sleep issues. He has yet to present for further services.

11 Treatment Implications of the Case

This case study provided an example of how CBT-I can be effective for patients with complex neurological conditions when flexibility is applied within the treatment protocol. Flexibility to the treatment as designed was imperative to meet Eric’s needs and physical constraints. Importantly, the protocol modifications were made thoughtfully so that fidelity to the CBT-I protocol could be as closely maintained as possible. Specifically, given that Eric’s time spent in bed each night was largely out of his control as his home aides were responsible for helping him to transfer into bed, creativity was required to implement the stimulus control portion of the treatment. The therapists worked collaboratively with Eric to develop a plan that included modifying the incline of his adjust- able bed to help discriminate between states of wakefulness and sleep. This helped to empower Eric to be active in his insomnia treatment despite his inability to ambulate from his bed until his aid arrived in the morning. In addition, given the lack of focus on sleep disturbance among patients with neurological conditions, the psychoeducation component of treatment was critical in helping Eric to better understand healthy sleep hygiene and to better recognize and understand his sleep patterns. Although he acknowledged having long-standing difficulties with sleep, these concerns had never been directly addressed with him from a behavioral standpoint. Eric was eager to engage in the treatment and adhered to his homework assignments—another critical driver in the success of his treatment that highlights the ability for successful engagement in this treatment despite the complexity of his medical and neurological conditions. By the end of the treatment, Eric was able to more independently manage his bedtime routine and implement relaxation strategies to help ease his nighttime anxiety. Overall, his success in treatment was highlighted most by the collaboration he and the therapists used to modify the treatment protocol to best meet his needs, which, in turn, allowed him to be optimally and actively engaged in his treatment.

This case study highlights the success of making modifications to the CBT-I protocol to treat insomnia in a patient with complex neurological challenges. However, the outcome of this par- ticular patient’s treatment should not be overgeneralized as neurological difficulties are complex and nuanced. To this end, the modified CBT-I treatment may have been successful for many reasons. One reason may be the creative adjustments made to the protocol, such as the stimulus control modifications and adapted bedtime routine. It could also be that the therapeutic alliance helped Eric to feel that his sleep difficulties were acknowledged and understood and that he was able to take control and make positive changes, thereby gaining a sense of self-efficacy. While it is beyond the scope of this article to warrant support for any one of these hypotheses, this case does highlight the need for more rigorous and representative research within the area of insomnia treatment for patients with complex neurological histories, especially given the prevalence of sleep disturbance among this population (Pillai & Leverenz, 2017).

12 Recommendations to Clinicians and Students

Eric’s presentation required multiple modifications to the manualized CBT-I treatment, informed by previous research, indicating that flexibility with manualized protocol is at times beneficial

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and necessary. Because Eric has limited mobility and was largely confined during the day to his bed, his providers were required to adapt some of the behavioral recommendations, in particular, to maximize benefit. The prospect of making changes to an established protocol can be intimidat- ing to students and clinicians alike, particularly relative to the wish to maintain treatment fidelity and impact on potential outcomes. Mignogna et al. (2018) conducted a preliminary study that determined that a cohort of providers (N = 18) demonstrated consistent and appropriate fidelity to brief manualized CBT interventions in a primary care setting despite numerous adaptations to protocol. These providers indicated that adaptations to the protocol were made to better adapt to their patient populations based on the needs of both the patient and various systems, as well as the strength of the therapeutic relationship. They, ultimately, concluded that further research was needed to explore the possibility that fidelity to manualized interventions could exist on a spec- trum of adherence, potentially allowing for appropriate and effective modifications to meet patient needs.

With regard to the correlation between treatment adherence and outcomes, Webb et al. (2010) conducted a meta-analysis of 32 studies comparing treatment adherence and treatment outcomes, and ultimately concluded that variability in adherence did not significantly impact outcome. They cautioned, however, against the numerous potentially confounding factors in measuring therapy outcomes, some of which were observed in the Mignogna et al. study (e.g., therapist responsiveness, varying length of treatment). Later, Truijens et al. (2019) replicated Webb and colleague’s meta-analysis using 15 studies from 2010 forward, and their results also indicated that greater treatment adherence did not automatically link to greater effectiveness, though, like Webb and colleagues, they observed that an expansion of the research would be important to determine this with greater certainty. Exploration of adherence and treatment efficacy is far from having been exhausted, and there is ample room for clinician–scholars to make meaningful con- tributions to its literature.

The outcome of this particular intervention supports the idea that clinicians and students should consider the possibility that there will be important and appropriate ways to incorporate clinical judgment and creativity when adopting a manualized treatment approach. There may be opportunities to be flexible to adapt to the needs of each individual patient, while maintaining a solid grounding in and with reference to the research and theory that contributed to the creation of the intervention.

Appendix

Treatment Data

Table A1. Baseline Sleep Diary Data.

Week 1 Week 2

Bedtime 9:12 p.m. 9:27 p.m. Wake time 6:21 a.m. 6:18 a.m. Sleep latency 145 min 189 min WASO 240 min 170 min Early morning awakening 110 min 102 min Average time in bed 9.25 hr 9.48 hr Average total sleep time 2.69 hr 2.56 hr Sleep efficiency 29.04% 26.70%

Note. WASO = wakefulness periods after sleep onset.

112 Clinical Case Studies 19(2)

Table A2. Pre–Post Sleep Diary Data.

Week 1 Week 22

Bedtime 9:12 p.m. 11:00 p.m. Wake time 6:21 a.m. 6:42 a.m. Sleep latency 145 min 60 min WASO 240 min 60 min Early morning awakening 110 min 0 min Average time in bed 9.25 hr 7.86 hr Average total sleep time 2.69 hr 5.86 hr Sleep efficiency 29.04% 74.49%

Note. WASO = wakefulness periods after sleep onset.

Figure A1. Changes in Eric’s average time in bed and average sleep time across treatment sessions. a Denote appointment weeks.

Figure A2. Changes in Eric’s sleep efficiency across treatment sessions. a Denote appointment weeks.

Loveless et al. 113

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

ORCID iD

James P. Loveless https://orcid.org/0000-0002-4204-6221

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Author Biographies

James P. Loveless, PhD, MEd, NCC, is an assistant professor in the department of psychology at Middle Tennessee State University. His scholarly interests include cardiovascular psychophysiology, sleep, and predicting treatment adherence among those with chronic health conditions.

Jaclyn M. Russo, MA, MEd, is a doctoral candidate in clinical psychology at the University of Virginia. Her primary research interests include the ways in which neighborhood features influence the physical and men- tal health of children and adolescents. She is currently completing her pre-doctoral internship at the Ann and Robert H. Lurie Children’s Hospital of Chicago/Northwestern University Feinberg School of Medicine.

Virginia C. Andersen, PhD, is an assistant professor in the Department of Psychiatry and Neurobehavioral Sciences at the University of Virginia. Her scholarly interests include behavioral health in HIV positive populations, psychological outcomes post-transplant, and use of telemedicine to reach rural populations.