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Sleep Apnea Patient Centered Outcomes Network (SAPCON): Part 2

Last year, Sleep Review Magazine published an introductory story about the SAPCON project, which focuses on collecting input from patients with sleep apnea as a way to “democratize” research and gain valuable insights to promote more effective treatments.Sleep Review published a follow-up story to discuss the preliminary findings, which you can read about here on pages 26-28. 

This article is a continuation from Part 1 that covered the first challenge: They said I need a machine, that’s it?

Second Challenge: So I have sleep apnea? So what?

The summary provided by the authors outlines how patients may not be properly educated after diagnosis, focusing on three main gaps in understanding:

  • there is a relationship between sleep apnea and other co-occurring health problems,
  • treatment of sleep apnea may prevent or improve these comorbid problems, and
  • treatment may lead to overall health improvements.

Included quotes from patients confirmed they were not aware of the above, in addition to noting the lack of education on the “dangers of non-adherence.”

Two anecdotes showed how enhanced education leads to greater adherence. One patient reported his sleep doctor published a paper on the link between sleep and heart disease, which motivated the patient to use his PAP device whenever and wherever he would sleep. Another patient exclaimed she was never told why sleep apnea needed to be treated, and only when she learned about the connections to early dementia-like symptoms was she motivated to achieve compliance.

Many of the themes above are predicated on fear-based motivation, that is, the belief patients will move forward with treatment and maintain treatment compliance to prevent worse problems. The most motivation in such scenarios often revolves around heart conditions, because patients generally take heart disease more seriously than other illnesses, save the diagnosis of an acutely disabling condition or a terminal illness.

Heart symptoms quickly grab a person’s attention, because you can feel things like chest pain, palpitations or shortness of breath and notice difficulties in trying to exert yourself when suffering various cardiac symptoms such as arrhythmias, heart attacks, congestive heart failure and ischemic chest pain (angina). While not everyone responds the same way, a sizeable proportion of cardiac patients pay close attention to their heart symptoms, seek care for them, and demonstrate some eagerness in wanting to move forward to improve the outlook of their condition. Thus, patients with recent heart attacks or heart failure often show interest in learning about and treating sleep apnea if presented in a balanced fashion.

One caveat relates to the frequent use of supplemental oxygen during cardiac hospitalizations. Regardless of the type of mask (nasal prongs or face mask), a majority of cardiac patients report annoyance with needing to sleep with something on their face, which can evoke unpleasant memories when first confronting PAP therapy. Moreover, many heart patients suffer emotionally distressing experiences during acute cardiac illness (like believing or feeling like they might die), and these unpleasant experiences often arose during labored breathing or shortness of breath. Thus, a PAP mask, headgear, tubes and the machine may not immediately resonate with a cardiac patient, if they had developed a claustrophobic-like response in the hospital environment. A balanced approach may require starting with the unobtrusive nasal pillow masks (even if the patient mouth breathes) to avoid feelings of claustrophobia in a recently “traumatized” heart patient.

Fear-based motivation, however, does not always work in variety of circumstances where the pain and suffering seems improbable and in the distant future. If you informed an OSA/UARS patient “you will be a safer driver and suffer fewer car accidents,” the information is likely to not register in someone who already defines himself as a careful driver and whose had no driving mishaps in the past decade. A similar disconnect might arise in telling someone that treating sleep apnea might decrease the risk of hypertension, but perhaps the patient only currently knows of a family history of high blood pressure and her most recent readings were normal. Thus, specific clinical relevance to the patient is the more likely path for success. Even a minor condition that irritates a patient produces more motivation.

The most common experiences we see with risk-reward scenarios occur in patients who suffer both snoring and nocturia. Which one do you suppose leads to greater motivation to adhere to PAP therapy? The key distinction between these two symptoms is based on the state of consciousness when they occur: you snore while asleep; you get up to visit the bathroom while awake. Therefore, nocturia is the more vexing symptom to the patient; whereas, snoring is more annoying to the bed partner.

The impact of nocturia may go beyond annoying. Suppose you suffer from insomnia; then, nocturia will worsen the problem of unwanted bouts of sleeplessness. In this circumstance of comorbid conditions, the patient recognizes the direct and immediate opportunity to decrease insomnia—a very large reward in the eyes of any insomniac. While the individual may still be in the frame of mind to prevent something—insomnia—the typical insomniac is usually not afraid of insomnia, albeit such fears do arise in more severe cases. Then again, you could argue a patient is motivated by the fear of having a bad night of insomnia after a nocturia event, when confronted by a particular set of important next-day activities. All these aspects may serve to motivate an insomniac suffering from nocturia to adhere to PAP.

As another example, suppose you are elderly or physically disabled and are concerned about the risks of falling and injuring yourself in the middle of the night. Now, there is a real fear each night as navigating safely in the bedroom requires considerable attention. This fear seems more substantial in that falls at night in the elderly are a major cause of broken hips, prolonged recovery and rehabilitation, and directly or indirectly lead to death in the first year after the fall in a significant percentage of cases (ranging from 14 to 58% in different types of patients).

By way of comparison, which set of symptoms, insomnia and nocturia or heart attacks and hypertension, do you suppose would prove more motivating for patients? The answer largely follows the degree of immediacy versus the degree of danger as described in the samples above. Someone highly annoyed by awakenings or trips to the bathroom at night will want to pursue immediate treatment steps; whereas, someone with a recent heart attack or hypertensive crisis may be highly motivated to pursue PAP to prevent deadly consequences.

In clinical practice, it is remarkable how these scenarios play out, especially with individuals who do not report many sleep complaints. In this common scenario, the patient might report very little in the way of medical co-morbidity, not even high blood pressure, but his physician referred him for sleep testing because antidepressants had not worked to improve his daytime energy level or fatigue. Unless the fatigue is fairly severe and the energy level noticeably low, the patient is much more likely to forego any further treatments for OSA/UARS, because he would describe the cure as worse than the disease. Moreover, denial is easy when there isn’t much to deny: if the level of fatigue or low energy does not seem to markedly affect the individual’s lifestyle, the person might respond by declaring he’ll drink one more cup of coffee in the afternoon as the best solution to his energy problem.

Patients with greater sensitivities toward their mental functioning are more likely to be interested in treatment, because they notice decrements in memory and concentration. Others who pay close attention to physical fitness may be motivated by the potential for a renewal of an exercise routine. But, if someone wrote off either of these conditions to aging or simply chose not to complain about cognitive dysfunction or about their lack of physical fitness, why would they pursue treatment for OSA/UARS?

Teasing apart symptoms that patients primarily complain about is usually the most direct pathway to engage them to ramp up PAP therapy use, and this approach often proves more expedient than relying strictly on the general, fear-based education model.

Sleep Apnea Patient Centered Outcomes Network (SAPCON): Part 1

Last year, Sleep Review Magazine published in an introductory story about the SAPCON project, which focuses on collecting input from patients with sleep apnea as a way to “democratize” research and gain valuable insights to promote more effective treatments. Just last month, Sleep Review published a follow-up story to discuss some of the preliminary findings, which you can read about here on pages 26-28.

The article, which was written by three leaders of the project, reported initial information gathered through the www.MyApnea.org forum (the location of the SAPCON project), where patients described, “significant gaps in the care and information they receive from sleep medicine professionals.” With this background material, they sought additional feedback from users of the website via an extensive list of interesting SAPCON surveys that inquired about what patients see as information gaps, what they are missing from their clinicians, and their suggestions for best practices. The article then goes on to list a set of challenges and potential solutions for a professional audience. In the following posts, we’ll discuss each of the challenges and the solutions offered as well as how our sleep center, Maimonides Sleep Arts & Sciences, Ltd might offer additional or different approaches.

First Challenge: They said I need a machine, that’s it?

The essence of this complaint is the failure of healthcare providers to fully educate patients so they have a clear understanding of what it means to be diagnosed with sleep apnea. Patients are often unable to digest information about apneas or hypopneas (and no mention is made here of UARS, RERAs, or flow limitations). The main insight is that patients often find the learning process overwhelming due to different learning abilities and preferences. Additionally, sleep doctors are not taking enough time to educate their patients, or they lack specific plans or information sheets to spell out the process for patients to follow over time.

What surprised me most about this challenge point was the absence of any mention of underlying cognitive impairment in these patients due to their OSA-induced sleep deprivation or fragmentation. This impairment reflects our default position in dealing with every patient with a sleep disorder, because we know we must use great precision to tailor our educational and coaching pathways to effectively engage them. What I learned about this process in the 1990s was quite a surprise, because I had always assumed talking with a patient before a sleep study (the traditional approach to care) would prove a successful model of care. It never occurred to me most patients actually forget sizeable chunks of information following these appointments, especially when I was working at an institution where the patients could not be tested for a month or longer following the appointment. Although my research on this process point fits into the category of “straw poll,” it nevertheless was provided to me by a slew of sleep technologists who confirmed with me that most patients were not remembering much from their initial intake encounter.

When we opened our sleep center in 2003, we immediately moved in the direction of “shoot first, ask questions later.” The model comprised an extensive on-line intake system, which after completion by a patient led to phone discussions with my staff or me to clarify how to proceed. Because of the uncertainty on how patients might respond to the uniqueness of the system, a strategically placed question at the end of the Sleep Medicine History component of the intake offered the patient a choice of starting in clinic versus starting in the lab. At the inception of our program, I was stunned that greater than 80% of our patients opted for the sleep lab. As of today, greater than 95% of patients choose the sleep lab over a clinic appointment to start their sleep treatment program.

As you can see below, the wording is geared toward encouraging patients to attempt the sleep lab first, yet provides enough mentions of clinic appointments so that a patient would feel comfortable requesting one. If uncertain, this issue can be addressed over the phone to determine how to proceed. Occasionally patients who want a sleep lab test are persuaded to instead start with clinic and vice versa.

Visits to Dr. Krakow’s sleep medical center include clinic appointments and sleep lab tests. Although you may schedule clinic visits as you need, Dr. Krakow advises starting in the lab. Sleep tests not only clarify the nature of sleep problems much faster, but they also speed up your treatment program. In some instances, a patient benefits by starting in the clinic. If you are unsure about how to schedule, our staff will help you decide. (The patient checks the box by one of these two options.)

     I would be comfortable following your recommendations for scheduling.

     I would prefer starting with a clinic appointment to discuss my sleep problem

This lab-oriented system, however, served a much greater objective in attacking the cognitive impairment head-on through picture-focused education the morning after the sleep study. We recognized “strike while the iron is hot” as an essential ingredient to enhancing patient education and motivation. Thus, we created the following system:

  • Upon completion of the sleep study (diagnostic or titration), the patient sits down with the sleep technologist that morning for anywhere from 15 to 60 minutes to review images on the computer showing breathing events, leg jerks, arousal activity and so on and to discuss the importance of treating sleep-disordered breathing. Of course, the sleep technologist cannot make a diagnosis, but these preliminary visual impressions have proven very memorable to our patients and, in turn, informative and motivating.
  • Each sleep study report is generally finalized within 24 hours of testing, and greater than 90% are done the same morning, shortly after the procedure is concluded.
  • Our Results Coordinator (RC), using HIPAA-compliant, encrypted emails, sends the final report with my interpretations and recommendations to the patient (some patients walk-in for the report or have it faxed or mailed to them).
  • That same day, the RC phones the patient to discuss results and answer certain questions. In many instances, patients are so motivated by the process, they choose to go forward with a titration test as their next step or to request immediate set-up with PAP therapy.
  • If or when a patient expresses concerns, confusion, resistance, or impairment or any other potential barrier, the case is turned over to the Clinical Manager (a licensed, registered sleep technologist), after which the patient will be scheduled for either a clinic appointment or a phone call with me to determine the next best steps.

We also recently published our Patient Process brochure, detailing in 16 pages and largely with graphics, the sequence of steps from “ABCs to ZZZs.” The pamphlet is especially relevant for those patients who need frequent reminders about the process they are undergoing and for those who just want a one-time overview of how things likely unfold. The brochure, which will soon be available on our website (www.sleeptreatment.com) for easier access, spends the first six pages with simple flow charts to show the progression through our sleep center operations. Next, two pages discuss the role of DME companies and how resupply of equipment occurs. The second half of the brochure describes how to assess whether or not you are gaining an optimal response to PAP therapy, the role of retitrations in the sleep lab to achieve optimal results, how to take advantage of various types of follow-up appointments, alternative OSA/UARS treatments, a brief bio on my work in sleep medicine and an extensive glossary of sleep medical terminology, described in lay words. From a convenience standpoint, when patients open the brochure the left-hand page shows a broad overview flow chart of several key sleep treatment pathways, and the right-hand page shows every direct phone number for any administrative, clinical, or laboratory service.

Undoubtedly, aspects of our system might lead to a patient falling through the cracks, which regrettably occurs in every medical clinic setting, be it traditional or non-traditional. Nonetheless, we are very proactive in reminding patients we are just a phone call or email away, and we have been operating a web-based “ticketing system” to address patient needs for non-urgent issues. Finally, in the past year we instituted a no-appointment, walk-in system to rapidly review objective data downloads and measure changes in outcomes (e.g. sleepiness, insomnia, and nocturia) in order to evaluate progress or lack thereof in various types of individuals who may need more regular contact with our staff.

Summing up, the SAPCON project is a fantastic concept, which has the potential to be a real game changer by discovering flaws in the current practice models of sleep medical care. Like any information gathering system, whether more broad-based like SAPCON or narrowly tailored like a targeted research protocol, the output developed from the efforts is greatly influenced by the initial input, namely, the relevancy of the questions will greatly impact the relevancy of the answers received. As we continue to explore the other Challenges posed in the Sleep Review article and the preliminary solutions, we will take the opportunity to attempt to predict whether or not they are asking all the right questions, an objective rarely achieved in this type of project or even a research protocol. And, we will continue our discussion on how our sleep center operations may tackle these problems in different ways. We trust some of our commentary will be informative for the SAPCON group and other sleep medical center professionals.

Last, to be clear and fair, the SAPCON authors and their team who have put this grand, highly commendable scheme into actual practice can only report so much information in a brief report such as the Sleep Review article; in this context, then, they deserve the benefit of doubt when a piece of information may not appear in their comments due to these gross space limitations. Thus, any point made in my posts that might be read as a criticism or an omission regarding their work has no such intent. Rather, I would hope these posts will be viewed as informative and speculative commentary.

In the next post, we will look at the Second Challenge: So I have sleep apnea? So what?

Second Opinions and the Rise of REPAP: Part II

We attempted our first submission of the REPAP paper with a leading sleep journal, which summarily rejected the work for many of the reasons described in the previous post. The reviewers emphasized methodological weaknesses as well what in our opinion appeared to be obvious confusion about how sleep centers currently address CPAP failure.

One of the first complaints described in the rejection letter was at odds with what most people think or know about the practice of sleep medicine: “The practice of mask changes, mode and pressure changes are the usual treatment for patients who re-present to sleep clinic and are willing to retry CPAP.” No doubt the reviewer did not carefully review our work, because we explained our sample comprised second opinion patients who failed CPAP at their original sleep centers, yet from these patients’ perspectives there was no additional support or suggestions regarding changes in mask, modes or pressures. And, in treating a fair number of CPAP failure patients outside New Mexico who made the effort to travel to Albuquerque for a second opinion, the same complaints were registered in the general form of “no other options were discussed,” or “no other equipment changes were recommended for pressure delivery modes or pressure settings.” Some reported they never underwent a single titration study in the sleep lab.

Another claim from the reviewers was as follows: “The select patient population willing to undergo multiple repeat titrations and obtain a second opinion at a special sleep clinic do not represent the bulk of CPAP failures in the community;” however, both points are indefensible as virtually no second opinion research exists in the scientific literature. For all we know many CPAP failure cases would be extremely motivated to obtain second opinions if offered the opportunity. Likewise, once these individuals were educated on the underlying flaws in the use of continuous positive airway pressure (CPAP), they might express considerable motivation to pursue a retitration to test out dual-pressure technology.

A different reviewer made a very interesting comment on the number of retitrations completed by patients who ultimate used PAP therapy: “Those who had multiple titrations had a greater proportion of CPAP users than those who had a single titration. It is unclear whether it was the persistence of the patient and/or the physician that generated the multiple titrations. Were the patients who underwent multiple titrations more likely to be patients who were dedicated to the treatment and persisted, which would make them more likely to be users than those who had one titration or was it that the single titration was adequate.”

The reviewer made valid points, because our study was not a randomized controlled, prospective design in which we investigated one group undergoing single retitrations compared to another group undergoing multiple retitrations to determine which group showed more PAP use and adherence. However, at the same time the reviewer seems to neglect that every patient entered the REPAP protocol with virtually the same baseline, that is, attempted CPAP and either rejected it immediately or ceased use at some point after completing setup and trying to adapt at home. Therefore, any patient in our sample who became a user was deemed to have reversed CPAP failure, and those who renewed use were significantly more likely to have undergone more than one retitration study. Again, it’s a valid statement we did not prove that multiple retitrations were a key aspect of reversing CPAP failure, but nonetheless, common sense tells us that just by bringing these patients back to the sleep lab to try out dual-pressure, auto-adjusting technology and then have them report the experience as more comfortable and positive than past trials withCPAP strongly hints at the potential value of this model of care.

The most remarkable comment in this initial rejection of our manuscript was as follows: “There was a missed opportunity to emphasize the importance of evaluating the presence of residual events with the data provided by CPAP software rather than dwell on retitration as a dose adjustment. Unlike medication adjustments, the success of the titration for the most part, is quite evident during the procedure hitting the mark of AHI <5. Therefore, I would take exception for characterizing the current method as “one and done”. It is ‘one and done’ because it can be.”

The one and done remark refers to our introduction in the manuscript highlighting how many sleep centers only complete one titration for the vast majority of patients and then claim optimal results have been achieved because the AHI < 5. However, there are many discrepancies in the reviewer’s comments because the AASM standard for any titration is RDI < 5, because the RDI metric is much more accurate in measuring residual breathing events. For these reasons, and contrary to the reviewer, we believe PAP therapy is exactly like an adjustment to a medication dosage, and we would go so far as to exclaim that the standard of care should be to retitrate OSA/UARS patients in the first few months or years of use, because their pressure settings frequently need adjustment upwards or downwards to improve individual clinical responses. The bold statement by the reviewer implying that “one and done” is a very successful model has little if any support in the scientific literature. Rather, one and done is actually a conventional wisdom followed by a huge proportion of sleep technologists and specialists, because they have chosen to follow outdated guidelines (AHI instead of RDI) and perhaps are intimidated by their relationships with insurance carriers (spending healthcare dollars on retitrations).

Naturally, in any review there are useful pointers on how to improve the manuscript, but I trust you can see from the above criticisms, the reviewers demonstrated either a lack of knowledge on our approach to care or simply rejected our ideas as lacking clinical relevance. No surprise, then, the editor and reviewers rejected the manuscript.

The second attempt on the manuscript was submitted to another well-respected and widely read (by sleep professionals) sleep medicine journal, and the progression of events culminating in another rejection were very illuminating, because we chose to appeal their decision, which revealed even more gaps in knowledge regarding how some sleep experts manage CPAP failure.

In this appeal response to this second rejection from the second journal we noted: “Technology to our way of thinking is the elephant in the room as prior research routinely ignores the pervasive subjective complaints and objective findings of expiratory pressure intolerance.” In another words, the reviewers all but ignored or rejected the idea that technology might play an instrumental role in overcoming CPAP failure by addressing a physiological side-effect of CPAP.

A particularly telling comment along these lines emerged from a reviewer declaring our manuscript had “too much detail.” We could only assume this complaint was directed at our detailed discussion on the mechanics of expiratory pressure intolerance arising when a CPAP titration attempts to eliminate RERAs (flow limitations), the discrete and more subtle breathing events observed in exclusively in UARS or partially in OSA. By providing this detail, we had hoped to persuade reviewers a rationale for the application of advanced PAP technology to directly address expiratory pressure intolerance and thus outline a scientific hypothesis to test by using ABPAP or ASV in CPAP failure cases.

The next complaint, a fair one to be sure, was whether or not the REPAP protocol would be cost-prohibitive. The specific comment from a reviewer though again proved more telling, “For example, given the current climate of insurances severely restricting repeat titration studies one begins to wonder if retitration is the best form of action.” To our way of thinking, this conflation of insurance carrier restrictions somehow determining the best care for our patients may be understandable, but it still seems inappropriate. That is, regardless of cost, it is the physician’s duty to determine and recommend the best forms of treatment for patients. How the patient proceeds with this knowledge depends on many factors of which cost and insurance reimbursement are certainly relevant. However, the context of our research paper frames this issue in a patient-centric manner. We reviewed charts on individuals who rejected or ceased use of CPAP once started, which by all current evidence is an extraordinarily expensive state of affairs due to the innumerable health and economic costs linked to untreated UARS/OSA. By offering this perspective, our paper noted the importance of reversing CPAP failure in 70% of patients undergoing second opinions, thereby bringing into play a new set of health and economic factors likely to far outweigh the cost of one or two “extra” retitrations. For example, two retitrations might cost an insurance carrier around $1500 to $2,000 in New Mexico, but a hospitalization for a heart attack or a motor vehicle accident could easily exceed $50,000 or even $100,000.

A reviewer then made several interesting points about the degree of follow-up in this cohort of patients prior to their seeking care at our sleep center, the critical role of follow-up to improve adherence, and then raised the question of whether or not education, not technology, was the more relevant factor in the how and why of our patients reversing CPAP failure.

On the first point, our manuscript detailed how CPAP failure patients complained about numerous gaps in their follow-up at previous sleep centers, including the specific lack of attention to the potential use of new masks or PAP modes. Several other complaints were lodged with the process that directly applied to or indirectly hinted at technology failure such as pressure intolerance, struggling to adapt, mask discomfort and leak, and no perceived benefits. Regarding the second point on follow-up in general, the reviewer made no mention of a role for retitrating the patient in the sleep lab as if technology could not improve outcomes. Finally, in raising the question on the impact of education, we could only assume the reviewer believes that expiratory pressure intolerance is merely a psychological symptom that goes away once someone uses the device long enough. This conventional wisdom is rife within the sleep medicine community and in our view demonstrates a blind spot regarding the objective physiological stressors patients must endure to eventually adapt to a PAP device. We responded to the reviewer: “We find it difficult to imagine how education/insight from clinicians explains how patients overcame their discomfort with the PAP device and how it would have eliminated their expiratory pressure intolerance.” Clearly, because we routinely see ABPAP or ASV eliminating this pressure intolerance, we have chosen the path to treat failing CPAP patients more aggressively with this model of care; and, despite our chart review research (retrospective design), our data generate a testable hypothesis: can advanced PAP therapy reverse CPAP failure?

Last, one reviewer had considerable difficulty understanding our entire approach to the sleep lab as exemplified by this quote: “Moreover, it’s not clear to me how the method of re-titration described in this paper was developed. For example, were patients first re-titrated with CPAP and were switched to BPAP or ASV due to EPI? It’s also not clear what is meant by subjective and objective EPI. If BPAP was in fact needed, why use an auto BPAP instead of manually titrating BPAP?”

Subsequently, we attempted to respond to this critique, but the manuscript was again rejected, which prompted us to write a second appeal, particularly in light of this seeming resistance to how PAP technology might be delivered in a novel way. In this second appeal, we chose to introduce our response by directly attacking this issue in the following way:

“As a preface to our detailed letter, we wish to underscore a key issue that surfaces in every critique we have received from the reviewers. The issue involves the concept of manually titrating auto-adjusting, dual-pressure PAP technology in the sleep lab environment. Each reviewer in both versions of the manuscript expressed confusion about this idea or appeared to not understand the concept.

“The concept is straightforward. In the sleep lab, an auto-adjusting device can be set to auto mode, but then the attending sleep technologist manipulates all pressure settings on ABPAP or ASV devices to produce what amounts to a better response than the auto-mode only. Still, the auto-mode is retained, because the device continues to self-adjust to provide a better airflow signal, but through the added information of the manual titration, the pressure settings are now constrained in a more effective manner. Unfortunately, it appears our reviewers, like many other sleep researchers and clinicians, seem to only be aware of the use of auto-adjusting devices used with HST, but even then the auto mode may have only been used to set a fixed CPAP pressure.

“Our paper is describing how fixed CPAP or BPAP pressures cause iatrogenic problems to the airflow curve, which can be resolved by manually titrating auto-adjusting, dual-pressure PAP in the sleep lab.   The patient is then prescribed pressure modes and pressures according to settings determined in the sleep lab, based on the sleep tech’s titration and the physician’s interpretation.

“Thus, our use of auto-adjusting technology reflects a novel way to approach titrations and has no relationship whatsoever to the current use of auto-adjusting technology in the HST environment. We are spelling out this point, because it is clear that reviewers do not understand or are confused about what we are doing in the lab. And, we believe this lack of understanding or clarity has led to an inaccurate and incomplete review of our work.”

This second appeal was again rejected, prompting us to seek out another journal,Respiratory Care, which earlier in the year had given us a favorable response to our paper on adherence and sub-threshold adherence to PAP therapy when using advanced PAP technology. The publication of that paper prompted us to direct our next submission of the REPAP study to the journal Respiratory Care

Second Opinions and the Rise of REPAP: Part I

In prior work on this site and elsewhere we have put forth the necessity to use sleep lab retitration protocols as a mainstay of practice to solve the problem of CPAP failure. In this post, we will delve into the history on the development of this concept, which has culminated most recently in the acceptance of our first peer-reviewed, research paper on the REPAP approach (REPAP stands for repeat, rescue, retitrations to reverse CPAP failure). Our paper will appear online in the next few months in the journal Respiratory Care, after more than ten years of effort to bring to fruition.1

Our story begins in 2005 in the third year of operations at our new private sleep center,Maimonides Sleep Arts & Sciences. Initially, we were treating all patients with CPAP, auto-CPAP, and a few patients with CFLEX. With careful analysis of the airflow curve in the sleep lab, we eventually recognized a specific objective finding (described below) to explain the inferior results generated by fixed CPAP pressures. Going forward, we only used bilevel devices in the vast majority of patients. During this timeframe, we were already receiving an influx of patients seeking second opinions, and nearly all of them were using or had previously failed CPAP; these patients in particular guided us in narrowing our focus to explore the advantages of bilevel. The linchpin of all this work was the objective finding that CPAP patients could not tolerate fixed pressures when breathing out; bilevel alleviated the problem, providing considerable relief of both discomfort and distress. What surprised us most about our insights in 2005 and thereafter was the rarity of any discussion on the topic in the scientific literature. Specifically, we found few recommendations or strategies on how patients should be directed to proceed when they fail CPAP and want a second opinion.

In fact, we could only find a few articles on the potential value in conducting retitrations in some patients, but it was rarely spelled out that these patients were seeking second opinions. To this day, we are uncertain whether or not other papers or guidelines cover the concept of second opinions in the field of sleep medicine for failing CPAP patients. When you consider this omission from the scientific literature, we believe it should cause concern and possibly apprehension about the workings of the field of sleep medicine. After all, is it not a standard of care in virtually every aspect of medicine, surgery or psychiatry to educate and inform patients on the value of seeking second opinions for a host of medical, surgical and psychiatric disorders? I daresay virtually every person reading this post has either personally experienced the need for a second opinion or knows of a friend or family member who required one, if not more of these encounters to solve assorted health problems.

Why then are there no obvious tracking systems, research articles, or formal policy statements about second opinions in the field of sleep medicine? I believe no immediate or satisfactory answers to this question are forthcoming other than our field is relatively young compared to many specialties. Nevertheless, dispensing with diplomacy for a moment, I also wonder whether this lacuna is more representative of a large blind spot among sleep experts, a phenomenon likely to occur when a group of medical professionals form consensus opinions and policies about specific diseases or health issues, which then become set in stone. From my vantage point as something of an outsider who left the academic-oriented university environment and who has offered a number of criticisms of the American Academy of Sleep Medicine (an organization largely comprised of professionals with continuing ties to academia), I remain disconcerted by the lack of attention to second opinions in the field of sleep medicine.

In 2007, with this framework in mind, we began to collect cases from our database of patients who had come to us specifically seeking a second opinion, nearly all of them were using CPAP. As our caseload grew, we re-examined the scientific literature as well as policies and procedures from the American Academy of Sleep Medicine (AASM). To our surprise, there was almost nothing to read on the topic of second opinions except a few researchers describing how they had successfully switched patients from CPAP to auto-CPAP or bilevel. From the AASM the only salient point in their published procedures was the suggestion to consider bilevel in patients with complaints of expiratory pressure intolerance, usually due to high CPAP pressures defined as > 15 cm H20.

In 2008, we started writing our first draft of a manuscript to submit for peer-review publication. Our main intent at the time was to publish data on how switching patients from CPAP to BPAP not only improved comfort but also improved use and adherence rates. At this same point, we were also moving on from bilevel to auto-adjusting bilevel (ABPAP) devices and soon thereafter the even more advanced auto-bilevel device known as adaptive servo-ventilation (ASV). Because of this transition in the types of modes applied at our center, we paused temporarily in our manuscript preparation, because we were seeing better results with ABPAP and ASV compared to fixed BPAP.

Another issue that appeared, one that took a few years to iron out, involved how to define CPAP failure. By 2010, we had already seen more than 1000 second opinion patients, but with greater scrutiny we realized many CPAP failure cases actually represented various stages of noncompliance, which encouraged us to developed an internal “CPAP failure timeline.” For example, obviously someone who completely rejected CPAP before ever trying it or perhaps trying it once in the sleep lab during a titration is different than a patient who filled a prescription for a device, took it home and attempted to use it for a few months or longer. Still more differentiation could be found among patients using CPAP irregularly for a long time with fair results versus those using regularly for a long time with poor results. Then, some patients reported great early results for several months and then a steady deterioration in sleep caused by other sleep problems or simply a decline in the response to PAP.

You can imagine many other permutations, but there was a larger problem embedded within our efforts to define CPAP failure, which we soon realized would make things more difficult in trying to publish a research paper. The problem, which we noted around 2011, was most other sleep experts, clinicians or researchers were not investigating CPAP failure in this much detail. Because so many sleep professionals by this time were entranced and obsessed in dealing with Medicare’s definition of CPAP adherence, the compliance concept of use > 4 hrs per night for 70% of nights permeated medical judgment and decision-making in the treatment of OSA/UARS patients.

Consider the most obvious example: a patient reports sleeping with CPAP six hours per night for 6 nights per week, which by the way is the standard we strive for in our patients. However, the patient reports persisting symptoms of insomnia and nocturia at night and sleepiness and fatigue during the daytime. Did you know that among a fair number of sleep professionals, the first approach to this patient might assume the co-occurrence of some other health problem, sleep-related or otherwise? To be sure, nowadays many sleep specialists would want to review a data download of the patient’s CPAP use, but viewing these data and interpreting the findings often yield cognitive dissonance because of the following: many data downloads do not properly record the discrete breathing events (flow limitations) of UARS; elevated leak values are frequently deemed in the “normal” range; many sleep staff ignore the presence of central apneas if only 1 or 2 centrals per hour are recorded; and finally, even today, the majority of sleep physicians do not even consider the potential value of a retitration study as an important component of the work-up to determine the cause of the persistent daytime and nighttime symptoms.

Thus, from our vantage point of trying to “sell” this research to sleep professionals who might be unwilling to define CPAP failure with greater precision, we knew our efforts would be wasted unless we could unequivocally convince reviewers of the absolute certainty our patients had failed CPAP.

To be clear, we think someone using CPAP regularly for many hours per night yet reporting poor outcomes is experiencing CPAP failure. This scenario does not mean we are not attentive to other possible causes of the problems, for example, two of the most common co-occurring issues observed in these types of poor responders would be depression or leg jerks. In various ways, we look for both subjective and objective evidence for these conditions in a large proportion of second opinion, failing patients, because our sleep center specializes in the treatment of mental health patients with sleep disorders. Nevertheless, while these problems emerge as a key factor in CPAP failure in a small proportion of cases, actual CPAP failure caused by none other than “CPAP Failure” is in fact the leading cause of…you guessed it…CPAP failure!

Sorry, just trying to emphasize a point that remains largely ignored in our field, namely CPAP does not really work as well as many sleep professionals profess, and perceptions that mislead sleep docs into thinking CPAP works well are often based on their lack of experience in trying out other modes of PAP therapy in exactly the kinds of patients who report obvious residual symptoms.

By 2013, we treated more than 2000 second opinion patients and spent the next several months sorting them into different categories of CPAP failure developed using our timeline of failure. During this exploration, we realized a majority of patients were currently attempting CPAP but with either poor results or constant struggles to adapt or both. Because these patients might be using the device—at any level of use—we sensed it might be too difficult to explain their failure problems to sleep experts reviewing our research manuscript. To solve this possible confound, we selected only patients with the most extreme or obvious forms of CPAP failure, which included patients who originally underwent a titration at another sleep center, during which CPAP was attempted, but then:

  • – Rejected and refused CPAP setup
  • – Completed setup, attempted CPAP, but then ceased use entirely

In our minds, there was no confusion about such patients designated as CPAP failure, yet we continued to polish our manuscript during the next three years in ways that informed us of new barriers we would face in trying to publish a paper on the REPAP protocol. Over that time, we predicted that two large obstacles would hamper our efforts, and these barriers related to how other experts and clinicians in the field of sleep medicine would view our aggressive attempts to titrate RERAs as well as our use of the sleep lab to manually titrate the auto-adjusting technology of ABPAP and ASV. Despite our small attempts to head these problems off at the pass, we would learn later once we completed our first attempt at a submission that reviewers could not grasp or simply rejected both ideas about treating RERAs and manually titrating ABPAP and ASV.

Although our foresight was vindicated in the long-run, in the near term we decided to directly attack these two obstacle with projects that would ultimately strengthen the REPAP manuscript, one a very in-depth study on the neglect of RERAs manifesting in theJournal of Clinical Sleep Medicine (JCSM) and the other a brief commentary explaining several factors that prevented past researchers from demonstrating the value of auto-adjusting PAP modes.

In the first effort, we examined seven years-worth (2006 to 2012) of original research papers in JCSM (219 that provided objective data on sleep breathing disorders) to determine whether or not other sleep researchers were holding up the AASM standard to score RERAs on the diagnostic or titration studies. (2) In the study, we showed how few published papers were adhering to these AASM recommendations as only 36 of 219 paper reported RERAs, and only 25 of 219 included the RERAs in the respiratory disturbance index (RDI). Moreover, another sign of high quality research is whether or not a paper acknowledges important omissions in a study design or in results to provide insight to improve future investigations; but, in the 179 papers that did not reports RERAs or RDI data, 157 made no mention of their neglect in the limitation sections.2

These proportions (< 20% use of RERAs or RDI; >85% failing to acknowledge the omission) were striking and indicated very weak endorsement of AASM recommendations described between 1999 and 2005. That RERAs were ignored by leading researchers in the field of sleep medicine, despite all the evidence compiled by the AASM indicating the essential need to treat RERAs, demonstrated the vast majority of sleep researchers publishing in JCSM were working at sub-standard levels of care in the diagnosis and treatment of OSA/UARS. The publication of our paper led to a commentary on the topic by Dr. Nancy Collop who in nutshell called out Medicare for not addressing this issue accurately and stated that regardless of what name is given to these more subtle breathing events, they need to be scored and titrated.3 Last, in a follow-up commentary in response to Dr. Collop and another researcher’s letter to the editor,4 we reiterated how failing to attend to RERAs in a titration sleep study would most likely lead to residual daytime symptoms in most sleep-disordered breathing patients.5

The second effort was also a commentary, and it specifically addressed the work of Powell et al in 2012, “A Pilot Study Assessing Adherence to Auto-Bilevel following a Poor Initial Encounter with CPAP,”6 which claimed ABPAP was no better than CPAP in attempting to improve adherence in patients doing poorly with CPAP. This paper represented a perfect opportunity for us to highlight the flaws in this type of research, which employed only the auto-mode of therapy instead of manually titrating the device in the sleep lab. In the same volume of JCSM that published their paper, the editor and colleagues, Quan et al, authored a commentary about the Powell et al’s paper, concluding that routine use of an ABPAP device was not indicated,7 yet Quan et al completely ignore the two primary flaws in the study design: 1) the lack of manual titration of ABPAP; and 2) the failure to address residual RERAs. The most telling quote from Quan et al indicated a strong aversion to technology solving CPAP adherence problems: “Finally, such negative device trials [auto-adjusting devices, etc.] suggest that any improvement in PAP adherence rates will more likely occur by addressing social and psychological obstacles to usage rather than newer technical innovations in PAP delivery or interface design.”7

In response, our commentary published a few months later, “Driving on ‘auto’: Hands-on is more effective than Hands-free,” we explain the flawed premises in both Powell et al and Quan et al works.8 We highlighted the inappropriate use of auto-mode without sleep technologist intervention as poor quality because it does not meet standards of AASM titration guidelines. Further, we explained how this approach fails to titrate out RERAs and prevent expiratory pressure intolerance. Last, we explained only through a manual titration overriding the auto mode can such approaches achieve successful outcomes.

With our background papers published in 2012, we returned to the drawing board to prepare our best effort at a new manuscript to describe the REPAP process, and the next three years proved highly instructive as we went through various rejections and appeals in two leading sleep medicine journals before landing a home for the manuscript in a respiratory journal in 2016.1 That story will follow in the next post.

 

References

 

  1. Krakow B, Ulibarri VA, McIver ND, Yonemoto C, Tidler A, Obando JJ, Foley-Shea, MR, Ornelas J, Dawson SC. 2016. Reversal of CPAP Failure with the REPAP Retitration Protocol. Resp Care (in press).
  2. Krakow B, Krakow J, Ulibarri V, McIver N. 2014 Commentary on the Frequency and Accuracy of “RERA” and “RDI” Terms in the Journal of Clinical Sleep Medicine 2006 to 2012. Feb 15;10(2):121-4.
  3. Collop, N. Breathing related arousals: call them what you want, but please count them. J.Clin.Sleep Med. 2-15-2014;10:125-126.
  4. Masi AM. The lumpers and splitters paradox. J Clin Sleep Med 2014;10:701.
  5. Krakow B, Ulibarri VA, McIver ND. 2014. A RERA by any other name…Journal of Clinical Sleep Medicine. Jun 15;10(6):703-4.
  6. Powell ED, Gay PC, Ojile JM, Litinski M, Malhotra A. A pilot study assessing adherence to auto-bilevel following a poor initial encounter with CPAP. J Clin Sleep Med 2012;8:43-7.
  7. Quan SF, Awad KM, Budhiraja R, Parthasarathy S. The quest to improve CPAP adherence–PAP potpourri is not the answer. J Clin Sleep Med 2012;8:49-50.
  8. Krakow B, Ulibarri VA, Sanchez JN, Kikta S, McIver N, Melendrez D. 2012. Journal of Clinical Sleep Medicine Driving on “auto”: hands-on is more effective than hands-free. 8(3):343-4

Sleep Onset Insomnia and Advanced PAP Therapy

In prior posts, we discussed how treatment of sleep onset insomnia with PAP therapy showed mixed results and delved into our research on the potential for greater improvements using advanced PAP devices (ABPAP and ASV) in these patients. With the very recent publication of our research paper, “A Novel Therapy for Chronic Sleep-Onset Insomnia: A Retrospective, Nonrandomized Controlled Study of Auto-Adjusting, Dual-Level, Positive Airway Pressure Technology,” we can now dig much deeper into our study methods and the precise details of the results to understand the clinical relevance of this hypothesis-generating study. This term for the study refers to its design not being at the highest level of evidence, which would have been a randomized controlled trial comparing ASV or ABPAP to a CPAP device as one example. Our nonrandomized design means the claims we offer are not proof ASV or ABPAP effectively treats sleep onset or early insomnia; instead, our findings indicate a need for more research on these ideas, because a hypothesis or theory was developed in our work to explain why advanced PAP devices might treat this problem when other traditional modes of PAP (e.g. CPAP) demonstrated mixed results.

In developing our research protocol, we started with the premise to isolate a group of patients with severe sleep onset insomnia (SOI) as opposed to a group with a mixture of equal degrees of early, middle and late insomnia. Thus, the chief insomnia complaint of eligible patients needed to be severe SOI, rated in two different ways. First, they needed to self-report sleep onset latency at bedtime greater than 60 minutes, and second on the Insomnia Severity Index rating system for question number one, they had to rate their difficulty in falling asleep as severe or very severe. By focusing only on these types of patients, we believed that whatever results we obtained would have more credibility because there would be no confusion about the type of insomnia we were attempting to treat with advanced PAP therapy.

Moreover, to solidify the perspective that these sleep onset insomnia patient were truly suffering from sleep onset problems, we asked them a series of questions that directly related to the most common symptoms seen in these types of patients. Here is a sample of a few of the behaviors indicative of sleep onset insomnia, and the percentages of patients who reported this issue:

  • – Racing thoughts keep me from sleeping (82%)
  • – Mind won’t turn off enough to fall asleep (72%)
  • – Clockwatching causes me frustration (67%)
  • – I clockwatch when trying to fall asleep (48%)

Of a possible 18 different indicators of insomnia in general and sleep onset insomnia in particular, each patient endorsed on average 9 of these maladaptive behaviors, and each patient averaged two or more psychiatric symptoms, conditions or disorders. All in all, our chart review collected 74 individuals with severe to very sleep onset insomnia whose profiles clearly established patterns consistent with the problem of early insomnia or SOI.

Other indications of severe global insomnia as well as severe sleep onset insomnia were demonstrated by their self-reported sleep patterns. They averaged less than 5 hours of total sleep per night despite remaining in bed on average for more than 8 hours, and they required more than 2 hours on average to fall asleep. They suffered from chronic insomnia for an average of more than 8 years, and 68% were using prescription sleeping pills, and 51% were using over-the-counter sleep aids. The sample included a mix of adult men and women, mostly White or Hispanic, average age 49 years-old, and mildly obese with an average BMI of 31 (roughly 25 to 30 pounds overweight).

Next, while all the patients were diagnosed with co-occurring OSA or UARS, we wanted to clarify why they were unable to tolerate standard CPAP therapy in order to provide the rationale demonstrating their need to attempt an advanced PAP therapy device. CPAP failure occurred in three phases of their treatment course:

Prescribed CPAP: 24 of the 74 patients initially used a CPAP device at home and failed primarily due to:

 

  • – Noncompliance (insufficient use)
  • – Subjective reports of difficulty breathing out (expiratory pressure intolerance,EPI)
  • – Poor outcomes defined as persisting insomnia, sleepiness or fatigue symptoms

 

Presleep/Desensitization: 48 of the 74 patients attempting CPAP in the sleep lab and prior to starting the actual titration failed CPAP during the desensitization trial primarily due to:

 

  • – Subjective reports of EPI
  • – Note: Actual instances of EPI equaled 65 due to patients failing on multiple devices (e.g. APAP, BPAP) before attempting advanced PAP

 

Titration: 69 of 74 patients titrated with CPAP, APAP, or BPAP in the sleep lab failed primarily due to:

 

  • – Needing variable pressure settings (auto-adjusting technology) to stabilize airflow signal
  • – Residual breathing events including central apneas
  • – Diagnosis of complex sleep apnea (residual central apneas meeting sufficient criteria)
  • – Note: Objective EPI equaled 83 instances, including failure on more than one PAP mode.

 

Within the 74 patients, the most striking difference was that some patients clearly were using their advanced PAP device more than others, which led to a dichotomy involving 56 patients who were using PAP on average more than 42 hours per week or greater than 6 hours per night (designated PAP Users) compared a Partial User group averaging less than 12 hours per week or less than 2 hours per night. Using this dichotomy, we compared how the severity of global insomnia and sleep onset insomnia changed in the two groups.

In the PAP User group, the Insomnia Severity Index total scores (ISI-TOT) dropped from 21.8 (consistent with severe global insomnia to 13.0 (consistent with less than moderate levels of insomnia severity). This change reflects what is known as a very large effect size, a statistical term that demonstrates the clinical relevance of a treatment response. In other words, a large effect size, or in this case a very large effect size usually coincides with a very large clinical change, which then is very noticeable to the patient. Small effects are usually less noticeable to patients while medium sized effects are noticeable but not as pronounced.

Remarkably, even the Partial User group demonstrated a large effect size with ISI-TOT scores dropping from 21.6 to 16.3. The changes between the two groups demonstrated statistical significance, which in this study is a way of declaring that the superior results in the PAP Users compared to the Partial Users was a very reliable finding, likely to be replicated if the research were repeated. However, to reiterate, the research is only a chart review and not a randomized controlled trial, the latter the highest form of evidence when attempting to prove the validity of a particular treatment.

The findings on the ISI-SOI single item score showed equally impressive results with the PAP User group demonstrating decreased ratings from midway between severe and very severe sleep onset difficulties to just under the moderate level of intensity. This change yielded a very large effect size double that observed in the Partial Users; yet even the latter group dropped their ratings from slightly more than severe to midway between moderate and severe. Again, both groups showed impressive improvements in sleep onset insomnia, but the PAP Users results were superior and statistically significant to the Partial Users.

An obvious question, one we could not answer in this study, was whether or not the two devices, ABPAP or ASV, were any different in their effects on SOI. The results were the same whether a patient used ABPAP (24 patients) or ASV (50 patients). And, regardless of the degree of use (PAP Users vs Partial Users), there were also no differences based on PAP mode. However, our findings on these points are not reliable given the numbers were relatively small when we divided up these patients by their status as Users vs Partial Users and by ABPAP vs ASV modes. A more sophisticated level of research design would be required to compare or contrast ABPAP and ASV effects.

One of the main clinical findings of the study was that many patients were using prescription sleeping pills or over-the-counter sleep aids, yet they were clearly failing these treatment approaches, and undoubtedly this failure explains at least part of their motivation to seek treatment at a sleep center. Moreover, their use of these medications combined with their strong endorsements of psychological factors as the primary cause of their sleep onset insomnia attests to the perspective that these individuals did not imagine a sleep breathing condition would prove to be an integral factor in their problems. The epitome of this mental framework was the ubiquitous problem of racing thoughts at bedtime. And, yet anecdotally, some patients reported a decrease in mental activation upon initiating and regularly use of advanced PAP therapy. These reports do not constitute evidence, but future studies must look carefully at whether or not racing thoughts are effectively decreased with advanced PAP. If so, such findings would strongly support a new theory on the underlying factors that cause racing thoughts as we have described previously, in which a psychological vantage point shifts to a physiological standpoint, albeit both could still play major roles in the development of sleep onset insomnia.

Along the same lines, there is no effort on our part to negate the potential role of medications or other forms of treatment for sleep onset difficulties such as sleep hygiene instructions or cognitive-behavioral therapy for insomnia (CBT-I). Rather, our work in this realm is about finding definitive or comprehensive treatment for various types of insomnia. We would be surprised if PAP therapy proves curative for a large proportion of patients with severe sleep onset difficulties, despite the provocative findings in our research. More commonly, sleep onset insomnia patients describe the problem of unfinished business when they hit the sack, which leads them to bring their work and worries into the bedroom and into bed.

If advanced PAP therapy ultimately proves a potent technique to decrease racing thoughts, then such information would be a game-changer in the treatment of insomnia. Nevertheless, it would still be likely such patients would benefit from other improvements in their coping skills to resist bringing unfinished business into the bedroom. These skills could be directly improved with sleep hygiene, CBT-I, and a host of psychotherapy methods. Finding comprehensive care for these patients is our primary goal, but we remain concerned that so many professionals in sleep medicine continue to overlook the potential relationships between OSA/UARS and chronic insomnia. We trust this new research will catalyze greater interest in more vigorous research protocols that use randomized controlled trials to learn how we can assist insomnia patients, and when relevant, how to treat that part of their insomnia properly addressed with advanced PAP therapy.