Obstructive Sleep Apnea and Adherence to Continuous Positive Airway Pressure (CPAP) Treatment: Let’s Talk about Partners!

Background: Continuous positive airway pressure (CPAP) is the gold standard treatment for obstructive sleep apnea (OSA). The benefits of this therapy were studied and analyzed over time; patient adherence is often poor, as many factors negatively influence it. A topic that needs clarification is whether adherence to CPAP treatment in a patient with OSA is influenced by the behavior of a partner or spouse. Methods: A scoping review was conducted to evaluate the role of partner involvement in the CPAP treatment management in a patient with OSA. The research project was performed between August and September 2021 by consulting the main biomedical databases: CINHAL, Embase, PsycINFO, and PubMed. Results: Among 21 articles considered valid for our aim, 15 are qualitative studies, 5 are quantitative and 1 presents a mixed method. We identified several thematic areas and “key” elements, which are prevalent in the studies evaluated. Conclusions: The presence of a partner promotes adherence to CPAP therapy in patients with OSA, resulting in ameliorating their overall quality of life. To increase CPAP adherence, a trained nurse could represent a reference figure to technically and emotionally support couples during the adaptation period and in the long term.


Introduction
Obstructive sleep apnea (OSA) is characterized by frequent complete (apneas), or partial (hypopneas), obstruction of the upper airway (UA) during sleep [1]. It is estimated that 2 to 4% of normal-weight individuals, 3 to 28% of overweight individuals, and 40% of obese individuals suffer from this disease, with a higher prevalence in men compared to women [2,3]. A small and collapsible UA, and a lack of responsiveness in the pharyngeal muscles during sleep, are some of the key factors responsible for OSA [4,5]. Thus, structural alterations of the UA, such as adenotonsillar hypertrophy or craniofacial alterations, may induce UA collapse. Increased muscle collapsibility might also be determined by adipose tissue deposits around the neck in obese subjects, or hormonal and muscle changes during menopause [6][7][8] and in older individuals [8].

Materials and Methods
We performed this scoping review according to the framework of Arkesey and O'Malley [43]. This framework consists of five steps: (1) identifying the research questions; (2) identifying relevant studies; (3) selecting studies; (4) extracting collected data; and (5) reporting results.

Identifying the Research Questions
To conduct this scoping review, the following research questions were identified: • Is partner involvement effective in CPAP management of someone with OSA?
• What strategies are implemented by couples to improve quality of life?

Identifying Relevant Studies
Starting from the research questions, search terms were defined using the population, concept, and context (PCC) format [44]: OSA patients; CPAP therapy; and CPAP adherence of the couple. The following databases were consulted: Cumulative Index to Nursing and Allied Health Literature (CINAHL), Excerpa Medica dataBASE (Embase), psychological information database (PsycINFO), and PubMed. The following terms were used, introduced either as free terms or as Medical Subject Headings (MeSH) terms: "obstructive sleep apnoea syndrome", "continuous positive airway pressure", "treatment adherence and compliance", and "spouses" (Table 1 search strategy). This was carried out independently by two reviewers, during the period between August and September 2021. ("Obstructive Sleep Apnea*" or "Obstructive Sleep ApnOea*" or OSAHS or "Sleep Apnea Hypopnea Syndrome" or "Sleep Apnoea Hypopnea Syndrome" or "Upper Airway Resistance Sleep Apnea Syndrome" or OSA or OSAS or "Obesity Hypoventilation Syndrome*") 5.
("caregiv* CARE GIVER*" or SPOUS* or HUSBAND* or WIFE or WIVES or PARTNER* or MARITAL or COUPLE or COUPLES) 6.

Selecting Relevant Studies
Studies were screened according to the PRISMA extension for scoping reviews (PRISMA-ScR) [45]. Two investigators completed the selection of studies after identifying and eliminating duplicate records using Mendeley. Inclusion criteria were studies describing the experiences of patients undergoing CPAP therapy for OSA, in English or Italian, patients aged 18 years or older, qualitative or quantitative primary studies, RCTs, observational studies, and published in the last ten years (2011-2021). Studies involving psychiatric and cognitively impaired patients were excluded.

Extracting Collected Data
All data were collected in a database. Data were extracted independently by two authors and classified into: author/year of publication; purpose; study design, sample, and methods; tools and strategies; results; and conclusion ( Table 2).  Semi-structured interviews, lasting from 12 to 60 min, were administered to the partners. Interviews were based on three open-ended questions, asking for a description of: a situation that facilitated treatment support; a situation that worsened the support; the management of these situations.
Five negative factors emerge: adverse effects, limited effect, practical and psychosocial problems, and inappropriate initial routine. Four positive factors emerge: effective treatment, improvement, motivation, and support. Three behaviors influence the partners' support in using the device: complete freedom (patient autonomy), shared management (supportive behaviour), and substitution (supervision and control).
The presence of the partner on the first days of treatment is a positive predictor of treatment adherence.
Henry & Rosenthal, 2013 [50] To illustrate the significance of the role and relationship with the partner in OSA patients, to diagnose, manage, and set up treatment.
A total of 10 patients (83%) report that the symptoms of OSA were identified by their partners: "my husband used to wake me up and say: Hey, you're not breathing". Male snoring is often considered "normal" compared to female snoring. Snoring is often mocked by the spouse in contrast to apnea. Patients often associate daytime symptoms with the disease. For 50% of the sample, body weight is the main cause of the problem. The willingness to tackle the problem is delayed. Love-hate relationship with CPAP. Three partners complain about the noise or intrusiveness of the machine, even though they are in favor of the treatment; three spouses, on the other hand, report relaxing because they no longer have to control their partner's breathing.
It is found that the role of the spouse is crucial in shaping problem identification, perception, help-seeking, and evaluation of the effectiveness of CPAP treatment in patients with OSA.   After randomization at 6 months, CPAP adherence and spouse involvement emerges only in the CPAP group (p = 0.01). After gender stratification, the association between spousal involvement and CPAP adherence is limited to men only, in a statistically significant manner (p = 0.03). Spousal relationship quality is not associated with treatment adherence. At three years, spousal involvement in the CPAP group is not associated with treatment adherence (p = 0.13), even after stratification by gender.
Spousal involvement is important in determining CPAP adherence during the initial treatment period, but has no effect on long-term adherence. Focus group (participants were divided into three groups). Breathing before the focus group.
The partners report the symptoms of OSA to the patient and remind them to perform the therapy. Symptoms are not noticed by most patients until treated with CPAP.
The key issue for patients and partners is the noise associated with air leaks and CPAP equipment. Overall feedback from participants is positive about both the effect of CPAP and routine adoption of therapy. All participants report improved quality of life.
The key role of the partner in identifying the symptoms (and variation) of OSA, and in the correct use of devices, emerges. The presence of family members is found to be the cause of the identification of the health problem ("for years and years, I didn't see it as a big problem, because I never counted apneas"); they are also credited with the success of treatment adherence.
The study led to the construction of a theory based on negotiating the positive and negative aspects of CPAP, and the balance of living with it. Partners are an integral part of the CPAP process, and should be included in the pathway from diagnosis to treatment management at home. The active presence of partners promotes understanding of the pathology, purpose, and management of therapy.   The partners also report improved values on all three scales from baseline to 3 months.
An educational and supportive intervention aimed at new CPAP users and their partners is feasible and beneficial. Improvements in sleep quality, daytime sleepiness, and daytime function are evident for both patients and partners in the CES group.
In the CES group, there is an increase in CPAP use in the first month, and a decrease at 3 months. The authors recommend further educational sessions. All partners report that the intervention helped them to support the patient in using the CPAP. This involvement may decrease with time. Tools: adherence to therapy → ECR; sleep quality → PSQI; daytime sleepiness → ESS; depressive symptoms and anxiety symptoms → PHQ-9 and GAD-7; relationship satisfaction → CSI-16 Adherence to CPAP treatment was recorded on a memory card in the CPAP machines (between T1 and T2).
There is no significant difference in the perception of anxiety between those who started the treatment prompted by their partner or autonomously (p = 0.049), and those who decided in agreement with their partner or autonomously (p = 0.04). Adherence to CPAP increases at T2, associated with increased use of the device (p = 0.02). Between T1 and T2, improved sleep quality and reduced levels of depression (p < 0.001) and anxiety (p < 0.001). No statistically significant difference between T1 and T2 in CSI (p = 0.26) The use of CPAP makes a significant difference in the treatment of sleep, a reduction in depression and anxiety at 1 month after treatment, but these are not sustained after 3 months of treatment.

Results
The following main focuses are identified: partner's engagement, anxiety and depressive symptoms, daytime sleepiness and sleep quality, quality of sexual life and marital relationship empowerment, facilitators, and barriers to CPAP treatment.

Partner's Engagement
Partner's attitudes influencing patient's device use can be classified as: (i) complete freedom ("patient autonomy"); (ii) shared management of the device ("supportive be-haviour"); and (iii) complete substitution ("supervision and control") [36]. "Supportive behaviour" consists of sharing CPAP management with the patient, by giving practice support and paying attention to the patient's needs. This attitude also consists of helping with device starting, putting the mask on in the evening, adjusting the mask during the night when air leaks occur, and, generally, being present to manage problems [36,62].
Being supported and providing support allows patients and partners to become a "team." Identification as a "we", independent of CPAP use [58], increases the motivation for CPAP adherence [36,38,48]. A partner's emotional support is associated with cooperation in treatment management within 3 months of beginning CPAP treatment [48]. This cooperation is particularly advantageous in reacting to a reduction in CPAP use [48]. Thus, in males and females married or cohabiting for more than one year, collaborative spouse involvement provides support and encouragement, and facilitates problem solving, with a positive impact on CPAP adherence [46,53]. In contrast, unmarried patients show greater ability and autonomy in managing the device by themselves [53]. In addition, the presence of other familiar components, such as children or grandchildren, appears to be crucial for CPAP adherence [23,38], and facilitates the establishment of proactive dynamics [53].
While educational and supportive interventions for both CPAP users and their partners during the first month of treatment are shown to increase adherence, this beneficial effect is not confirmed from 1 to 3 months, when only 1.6 h of CPAP compliance is reported [29]. After three years of CPAP use, spousal involvement seems not to be associated with treatment adherence, regardless of gender [55]. It is well known in the literature that adherence in the first month of therapy is a solid predictor of long-term use [30]. Thus, the positive involvement of the spouse at the beginning of CPAP use also determines long-term positive behaviour [30].

Anxiety and Depressive Symptoms
Increased energy levels and an improved quality of life, as consequences of OSA treatment, permit patients and their partners to re-engage in social and recreational activities [38]. Patients report that they do not feel tired during the day, and consequently, they feel happier and less irritable [38]. Moreover, the benefits observed over time from CPAP therapy increases patients' desire to live longer [23].

Daytime Sleepiness and Sleep Quality
The Pittsburgh Sleep Quality Index (PSQI) [63], the Functional Outcomes of Sleep Questionnaire (FOSQ-10) [64], and the Epworth Sleepiness Scale (ESS) [65] are used to assess sleep quality, daytime sleepiness, and its impact on activities of daily living, respectively. Improvements in PSQI scores are reported in OSA patients after one month of CPAP treatment [60]. When education and support is provided to both the patient and their partner [29], the FOSQ-10 improves after 3 months of therapy, together with a decrease in daytime sleepiness [54]. The benefit of CPAP treatment on daytime sleepiness assessed with ESS at 3 months also results in a better relationship [29,60] and an overall improved quality of life [29,60].

Quality of Sexual Life and Marital Relationship
Among the consequences of OSA, sexual dysfunction has an emotional impact on patients, with significant effects on mood and social relationships [52]. In partners, sexual performance is described as unsatisfactory at the time of OSA diagnosis, with significant improvements after OSA treatment [52]. In male patients, OSA is typically associated with sexual dysfunction [51]. After 12 months of treatment, a beneficial effect is observed in terms of erection quality, ejaculation time, sexual desire [51], and improved sex life, more so in men than in women [52]. Furthermore, among males, marriage quality, as measured by the Dyadic Adjustment Scale (DAS) [23,46,54,55,59], is positively associated with CPAP adherence [55]. In contrast, in women, OSA often leads to psychological difficulties [47], and treatment of OSA for 12 months is associated with improvements in sex life quality [47,51]. In women with OSA, CPAP treatment does not adversely affect partner relationships [47].
Independent of gender, spousal involvement increases CPAP adherence after 6 months [55]. However, after 3 years of treatment, spousal involvement in the treatment group is no longer associated with adherence to therapy, even after stratification by gender [55].

Encouraging Empowerment
Empowerment and competence are identified as positive predictors of CPAP therapy, and are crucial for patients' well-being and self-esteem [53]. Especially during the initial phase of CPAP treatment, the partner can help and support patient autonomy by improving spousal autonomy, sharing treatment management, and completing care of treatment management ("complete substitution") [49].
An autonomy-promoting attitude in a partner increases the use of CPAP in OSA patients [53]. In fact, from data analyses from device use time, individuals with high autonomy support use the CPAP for an average of 166 min more than patients with low practical and emotional support [53].

Facilitators to CPAP Treatment
Encouraging partners with key attitudes, such as giving support in the practical management of therapy, using verbal encouragement for device use, and using humor to manage the situation, are considered relevant facilitators of increasing CPAP adherence [53]. A partner's support and encouragement help the patient to feel less embarrassed, and consequently improves treatment adherence [36]. Positive factors for CPAP use include: (1) effective treatment and improvement of symptoms; (2) motivation; and (3) support from experts [49]. Couples believe that open, frequent, and supportive communication emphasizes and facilitates treatment adaptation [36].
Two studies report the involvement of experienced nurses [28,49]. In the first study [28], the nurse performs treatment education, explaining the fitting to all patients before the use of CPAP, follows up with the patients during the first week of treatment, and has follow-up visits at 3 months, 6 months, and then every 6 months [28]. In another study [49], experienced nurses provide information on OSA and adverse effects to both patients and partners. Couples report the need for support and assistance from healthcare professionals, which are perceived as an irreplaceable source of help and comfort [49].

Barriers to CPAP Treatment
Negative predictors of CPAP adherence include adverse effects, restriction of movement in bed, practical and psychosocial problems (e.g., difficulty in communicating with the mask), an inappropriate initial routine [49], feeling embarrassed or 'unattractive' in the eyes of the partner, and reduced adherence [36]. Furthermore, the 'supervision and control' of the partner is an attitude often judged as authoritarian, because of the imposition of directives that imply opposite reactions [36,62]. However, insistence on encouraging treatment, as well as derision, is not helpful for autonomy [53]. Popular culture and funny representations of snoring negatively impact adherence to CPAP treatment. Sometimes, men consider their wives' concerns about their "not normal" snoring to be excessive. Conversely, some women report that their snoring is comically interpreted by their spouses, making the situation difficult and embarrassing, even to the point of not being able to discuss it seriously [36].
Negative feelings, such as 'fear' or 'dread,' explain why the diagnosis of OSA is often delayed [36]. Dying while sleeping due to prolonged apnea is often an erroneous fear that patients' partners report and ask clinicians about during visits for sleep breathing disorders [36]. Anxiety is the predominant feeling at the beginning of treatment [53], which diminishes when the partner is supportive [35] and after CPAP treatment [36,60]. CPAP also seems to have a positive impact on depressive symptoms [36].

Discussion
The aim of this review was to assess the effectiveness of partner involvement in the CPAP management of OSA patients, and to analyze the strategies implemented by the couple to improve their quality of life. This scoping review identified the following themes: partner's engagement, anxiety and depressive symptoms, quality of sexual life and marital relationship, encouraging empowerment, facilitators, and barriers to CPAP treatment.
Patients develop a 'love-hate' relationship with CPAP. In some interviews, patients often report that the first morning after treatment was the best day of their life [50]. Despite this, studies indicate variable adherence; thus, more than half (56%) of patients give up using the device within the first month [66]. Our review indicates that the presence of a companion who is emotionally supportive and who, through positive behaviour, encourages the patient to carry out therapy is fundamental in creating a bond of loyalty between the device and the patient. This is because, in patients with chronic diseases, it is essential that the therapy is carried out in a consistent manner [41].
Healthcare workers involved in sleep medicine also need to highlight the role of a supportive partner in motivating adherence at an early stage. Therefore, the sleep team should share information with patients and partners at the early stages of OSA diagnosis, in order to enable the beginning of more informed treatment, and to enable better adherence and greater motivation not to abandon it. Furthermore, it is important for healthcare professionals to assess whether a partner engages in pushy behaviors that compromise adherence [36].
Partner support should be maintained long-term to promote adaptation to the device, increased rest, and a reduction in anxiety and depressive symptoms, resulting in an improvement in the couple's relationship and sexual satisfaction [47]. Recent studies show that the reduction of OSA episodes significantly improves daytime alertness [21], and both patients and their partners show an improvement in the couple's quality of life.
From the analyzed studies, it emerges that an experienced nurse is considered by couples to be a strong point within the team [28,49]. Thus, the inclusion of a nursing figure could provide support to both the patient and the partner, as it is often not easy to maintain a high level of motivation. Indeed, after three years, the involvement of the spouse no longer seems associated with treatment adherence [55]. This may indicate the need for specialist nurses to provide long-term educational interventions. The nurse could act as a bridge between the sleep physicians and the couple, identifying difficulties and referring patients to different specialists, who could also help improve long-term motivation. This would be in line with the creation of personalized and supportive pathways for precision medicine in long-term interventions.
The literature still lacks knowledge on the link among treatment adherence, the patient-partner dyad, and the phenomenon of mutuality. While mutuality was investigated in several chronic pathologies [39,40,42], there is a need for studies on OSA patients with longer follow-ups. In addition, it would be useful to evaluate the effectiveness of various types of educational interventions, as well as motivational interviews carried out by experienced personnel. Furthermore, it would be useful to analyze data on adherence and mutuality within LGBTQ couples, to determine if there are differences compared to traditional partnerships. The authors of this review also propose mixed method studies investigating the role of the nurse as a facilitator of adherence, or as a substitute if the partner is not present or can take over this role.

Study Limitations
The main limitations that were found in most studies are: the lack of sample assessment prior to CPAP treatment; a short time to determine long-term effectiveness on patients' health; the use of different assessment tools but assessing the same outcomes (e.g., anxiousdepressive symptoms, quality of life, and sleep-rest quality); small sample size; and no data were found on LGBTQ couples, which did not allow us to explain whether there are differences compared to traditional partnerships.

Conclusions
The findings of this review suggest that teamwork within the couple, allows the creation of a virtuous circle, and results in improved adherence to CPAP in OSA patients. Therefore, it is important for the sleep medicine team to be aware of the importance of the partner's role in promoting CPAP adherence. An experienced nurse could act as a bridge between the sleep physicians and the couple [67,68], identifying difficulties and referring the patient to different specialists to improve long-term motivation [69]. This would pave the way for personalized and supportive pathways in OSA treatments.
Author Contributions: Conceptualization, D.R. and E.R.; methodology, D.R. and E.R.; data curation, E.R. and M.F.; writing-original draft preparation, D.R. and E.R.; writing-review and editing, E.P. and A.L.; supervision, C.A., P.R., C.L. and G.P. All authors have read and agreed to the published version of the manuscript.