Introduction

Oropharyngeal Dysphagia (OD) is a clinical condition that implies abnormalities in the physiology of oropharyngeal swallowing. Swallowing safety (i.e. transfer of the bolus from the mouth to the esophagus without penetration or aspiration into the airway) and/or efficiency (i.e. transfer of the bolus from the mouth to the esophagus without residue) might be impaired. OD may be associated with life-threatening complications, such as aspiration pneumonia, malnutrition and/or dehydration [1–3]. OD prevalence in the general population amounts to 12.1% [4], while it shows ample variations in persons with stroke (8.1–80%), Parkinson’s disease (11–81%), traumatic brain injury (27–30%), community-acquired pneumonia (91.7%) [5], and head and neck cancer (HNC; 66% at diagnosis) [6].

Patients with OD may suffer from mild difficulties to severe inability to swallow, requiring bolus modifications and alternative feeding methods (i.e. nasogastric tube, gastrostomy tube) [7,8].

OD severity, diet restrictions to prevent complications, as well as related behavioral and social limitations may lead patients to experience depression, isolation [9], and decreased health-related quality of life (HRQOL) [10,11]. Changes in the meaning and experience associated with eating can also negatively impact on patients’ social roles [12].

Family members and friends of persons with OD may also be negatively affected by the condition, since they are frequently involved in the care process as informal caregivers, a term referring to individuals who provide practical and/or emotional support without payment [13]. Regardless of OD etiology, caring for persons with OD may result in remarkable physical and emotional burden [14–20].

OD clinical characteristics and impact on patients’ and caregivers’ lives were both investigated through primary evidence and synthetized by means of literature reviews. In particular, reviews targeted the management of OD in acute and critical care [21,22] and in nursing homes [23], the production of suitable food using texture modification technologies [24], the impact of OD and modified-texture diets on patients’ HRQOL [10,11], and the burden perceived by caregivers [25].

The physical and psycho-social consequences of OD for both patients and their informal caregivers are conducive to emerging needs. In motivational terms, needs refer to “the presence of a particular desire or preference, often rooted in a deficit or shortage, with such preferences varying widely between individuals” [26]. In the medical context, they comprise any necessity arising in the physical, practical, informational, social, psychological, and spiritual domains during the diagnostic, treatment, and follow-up phases of a disease [27], and requiring health professionals’ assessment and intervention.

To date, most efforts to address patients’ and caregivers’ needs consist of frameworks developed to increase efficiency and equity of healthcare services, taking into account organizational and contextual factors hindering or facilitating service access and use. Examples of this approach are the global WHO Framework on integrated people-centered health services (IPCHS) [28] and other more specific frameworks contextualized in community and palliative care [29–32]. In all these frameworks, however, the unit of analysis is the healthcare service rather than the person. The Supportive Care Framework (SCF) [27] attempts to classify individual needs of patients diagnosed with cancer along the different stages of disease through appropriate and timely services. The needs of persons with cancer were classified into seven domains: physical, emotional, practical, informational, social, psychological, and spiritual. Over the decades, the SCF was fruitfully applied to other clinical conditions such as Amyotrophic Lateral Sclerosis [33] and stroke [34]. To the best of our knowledge, however, it has never been used to investigate and classify the needs of patients with OD and their informal caregivers.

Another relevant aspect yet to be explored among patients with OD and their caregivers is the variety of needs-driven coping responses that shape their behavioral and psychological adaptation to OD. To this purpose, the Common-Sense Model of Self-Regulation (CSM) [35,36] may represent a useful framework. According to the CSM, when facing or anticipating a health threat (illness) individuals develop personal beliefs about both the threat (illness representations) and the strategies required to manage it (treatment representations). These representations lead them to identify and implement action plans to cope with the situation. Multiple sources contribute to shape illness and treatment beliefs: somatic sensations and deviations from normal daily functioning; prototypes and subjective representations of healthy functioning, illness and available treatments; opinions from experts and lay people, particularly family and friends. In the CSM, illness and treatment representations are articulated into five components: label and associated symptoms (identity); causes; course and duration (i.e. acute, chronic or cyclic timeline); expected effects on the individual’s life (consequences); availability of interventions to affect the illness course through personal actions (controllability) and medical treatments (curability). In addition, individuals form beliefs on their affective response (emotional representations). Several studies highlighted that the representations of illness, treatment and emotions affect health outcomes directly, as well as indirectly through the adoption of specific coping strategies (i.e. efforts to manage health threats and consequent emotions) [37]. Most importantly, this self-regulation system is shaped by the individuals themselves in a subjectively meaningful and comprehensible way. Therefore, individual representations of and expectations about illness and treatment may or may not be congruent with clinicians’ ones.

Based on these premises, the aim of the present study was to explore the literature investigating the care needs related to OD management of adult persons with OD and their informal caregivers during the last 20 years, using the SCF and the CSM as classification and interpretation framework respectively.

The following research question was formulated: What is known from the literature about the care needs related to OD management of adult persons with OD and their informal caregivers?.

We hypothesized that in the last 20 years the needs related to OD management of patients with OD and their informal caregivers (i) would cover physiological, behavioral, psychological and social aspects of patients’ and caregivers’ lives and (ii) would have been investigated mostly through qualitative research tools.

The decision to focus on the literature published in the last 20 years is related to the conceptual shift occurred at the beginning of the millennium in the understanding of disease and disability, leading researchers’ and practitioners to pay more attention to patients’ subjective perspective and illness experience. This shift clearly emerged in the International Classification of Functioning, Disability and Health (ICF), launched by the World Health Organization in 2001 [38] as a person-centered description of daily functioning, conceptualized as the interplay between body structures and functions, environmental features and personal factors. In the same years, the central role of patients’ illness experience was acknowledged in the Charter on Medical Professionalism [39] and in the SPIKES protocol, designed to support doctors in the stressful communication of bad news [40].

Materials and methods

The present scoping review complies with the Preferred Reporting Items for Systematic Reviews and Meta-Analysis extension for Scoping Reviews (PRISMA-ScR) [41] (S1 PRISMA checklist). Following the methodological framework proposed by Arksey and O’Malley [42], the study was articulated into five stages: identifying the research question; identifying relevant studies; study selection; charting the data; collating, summarizing and reporting the results.

A “best fit” Framework synthesis approach was adopted [43,44]. This augmentative and deductive method involves the identification of an a priori relevant theoretical framework against which to map data from included studies. At the same time, the integration of an inductive perspective was deemed as relevant, in order to allow for shaping the chosen framework on participants’ voices.

An operative protocol for conducting the review was agreed upon and shared by all team members, and registered on Open Science Framework (doi: 10.17605/OSF.IO/6ZCPK). The team was composed of seven members (3 speech and language pathologists, 1 phoniatrician, 2 psychologists, and 1 methodologist), contributing to the breadth and comprehensiveness of the review through their diversified expertise.

Results

Characteristics of the included studies

The search led to the identification of 2,881 records, to which 12 publications were added through manual search. After duplicate removal, 2,536 records were screened for eligibility by title and abstract, leading to the identification of 113 eligible publications. A careful reading of full texts led to the exclusion of 44 papers involving participants diagnosed with various pathologies, in which patients with OD could not be specifically identified. In addition, 34 papers were excluded because OD-related needs were not reported, 14 due to irretrievable full text, and 6 because of inadequate study design (e.g. case study) or insufficient information to extract meaningful data. Fifteen publications [21,50–63] were eventually included for data extraction. The study selection process is summarized in Fig 1 through a PRISMA flow diagram.

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Fig 1. PRISMA flow diagram summarizing the study selection process.

Adapted from Moher et al. [64].

https://doi.org/10.1371/journal.pone.0257683.g001

All the included studies were original works, 14 (93.3%) adopting a qualitative methodology and 1 (6.6%) quantitative methods. Data were collected through focus groups and interviews in the qualitative studies, and through a clinician-generated questionnaire in the quantitative study. Thirteen studies (86.6%) were published during the last 10 years, 7 of them (46.6%) during the last 5 years. All participants lived in Western countries, primarily English-speaking ones (11 studies, 73.3%). As usual in qualitative studies, sample sizes were rather small (participant n≤24), with one exception (n = 63) [58]. Most studies (10, 66.7%) were focused on patients’ needs, whereas only 5 (33.3%) investigated the caregivers’ perspective. The most frequent OD etiology (8 studies, 53.3%) was head and neck cancer; four studies (26.7%) involved participants with miscellaneous diagnoses and three other studies (20.0%) patients with neurodegenerative conditions. The characteristics of the studies included in the review are summarized in Table 1.

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Table 1. Characteristics of the included studies.

https://doi.org/10.1371/journal.pone.0257683.t001

The critical appraisal with the JBI tools [49] proved satisfactory for all the included studies, with the only exception of Brockbank et al’s [51] (S2 Table). In this study, the number of participants citing each need-related theme was not reported in the Results section, and conclusions were not clearly supported by the findings.

Needs categorization

A total of 266 supportive care needs were extracted from the 15 publications. Overall, patients and caregivers primarily reported social (N = 77; 28.9%), practical (N = 67; 25.2%), informational (N = 55; 20.7%), and psychological (N = 54; 20.3%) needs. Psychological needs were cited in 12 (80.0%) different studies, while informational, practical and social ones emerged from 10 (66.7%) studies. Physical needs were reported only by patients (13 citations, 4.9%) across 7 studies (46.7%). Spiritual needs were not reported in the studies selected for the present review. Table 2 shows the distribution of need categories, separately for patients and caregivers. The former mostly focused on the psychological, social, practical and informational domains, the latter on practical and social needs.

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Table 2. Patients’ and caregivers’ needs categorization.

https://doi.org/10.1371/journal.pone.0257683.t002

Table 3 shows the distribution of care needs in categories and subcategories, with exemplary quotations for each subcategory. Results derived from each publication and an audit trail of the coding process are available as additional material (S3 and S5 Tables).

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Table 3. OD-related needs classification with subcategories and exemplary citations.

https://doi.org/10.1371/journal.pone.0257683.t003

Within the physical domain, across clinical diagnoses patients expressed the need to consume tasty food and liquids [52,58,60] that could quench hunger and thirst, at the same time granting adequate nutritional status and swallowing safety [60,62,63]. They also expressed the need to attend SLP sessions, in order to learn techniques to improve their swallowing skills [55]. In the specific context of Motor Neuron Disease, participants relying on percutaneous endoscopic gastrostomy (PEG) as an alternative feeding method recalled their concern for body integrity preservation when they had been advised to undergo gastrostomy [57]. Patients with HNC, often experiencing mucositis as a consequence of radiotherapy, expressed the need to reduce pain while eating [52,63] and to adequately manage oral hygiene [63].

Patients and caregivers reported a wide range of practical needs, often formulated in terms of strategies and abilities allowing them to overcome difficulties. Two subcategories could be distinguished in this domain. Meal management included preparing food with adequate rheological and nutritional characteristics, assisting the patient in case of swallowing problems, investing additional time and thought on meals preparation and consumption, splitting lunch and dinner into small but frequent meals, and giving practical assistance to patients during meals (e.g. feeding the patient or reminding him/her of behavioral strategies for meal consumption) [18,52–54,56,62,65]. Food availability referred to the restricted choice of safe and tasty food in healthcare facilities, at home, or during social occasions [18,53,54,61,63].

As concerns informational needs, three subcategories could be identified: the need for getting additional, clearer, and tailored information. Patients and caregivers wished to acquire better knowledge about symptom development, PEG advantages, health professionals’ roles in OD management, and food recipes for OD [18,51–53,55–57,59,61,63]; they reported feeling overwhelmed by a large amount of information that was however unclear or difficult to understand [18,51,53,56,59]; they expected information to be more specifically tailored to their own experience [51,55,56,59,63].

Within Social needs, the most frequent subcategory was receiving support from caregivers or other family members, health professionals and other patients [18,51,55,56,61,63], followed by obtaining acceptance from family, friends, and community members [18,52,54,55,62,63,65], coping with changes in one’s family and social roles [55,56,65], and experiencing concerns for the other member of the dyad [53–56,65].

Among the psychological needs, the predominant one was to cope with the emotions evoked by eating difficulties, such as fear and anger, and with a feeling of loss [18,51,54–57,61,63,65]. Furthermore, patients and caregivers reported problem-focused [18,51,56,57,59] and meaning-focused coping strategies, such as acceptance of the swallowing problem as a part of one’s own identity, acceptance of a "new normal" condition, and recognition of changes in one’s own social roles [18,54,56,63]. In addition, patients and caregivers expressed the need to better connect the information received about symptom development with their own daily experience of OD [18,56,63]. Some patients reported the need to be motivated at eating, due to loss of eating desire [55,62,65], while others hoped to be able to enjoy food again [52,55,63]. Similarly, caregivers expressed the importance of maintaining a positive attitude in order to overcome eating-related difficulties [18,54] and to consider the caregiver role as an opportunity for personal growth [56].

Discussion

Overall, the present scoping review shed light on the dearth of research studies focused on the care needs of persons with OD and their informal caregivers.

A positive trend was nevertheless observed over time, as most of the retrieved studies were published in the last decade. As expected, data were primarily collected through a qualitative methodology, that offers the opportunity to delve into complex and underexplored phenomena such as unmet needs. On the other hand, the small sample size and the non-standardized tools for data collection, typical of qualitative studies, entail limitations in results generalizability and reproducibility [66]. These limitations must be taken into account when interpreting the findings emerged from the present review. The development and validation of a standardized tool to specifically assess the needs of patients with OD and their caregivers could contribute to enhance research rigor in this domain.

All the included publications demonstrated a satisfactory methodological quality, with the exception of one study, in which authors’ conclusions were only partially supported by participants’ voices. Only participants’ verbatim quotations reported in the Results section were however considered as units of analysis; therefore, we argue that the unsatisfactory critical appraisal emerging in this specific case would not impact the results of the review. In fact, drawing conclusions about the effectiveness of the interventions retrieved goes beyond the scope of the present review.

All the examined studies were conducted in Western countries. This finding may be related to one of the inclusion criteria, namely the use of English or Italian as publication language. It should be however noted that studies from Eastern Asian countries retrieved through the screening process had to be excluded due to their focus on OD treatment (e.g. acupuncture) rather than related needs [67,68]. It may be hypothesized that in these cultures patients’ and caregivers’ needs represent a rather private issue, not liable to investigation within the medical literature.

Over half of the included studies involved participants with a diagnosis of head and neck cancer. Such an over-representation may be related to the high relevance of swallowing disorders and related implications for these patients. OD may represent a less relevant concern for patients with neurodegenerative diseases and their caregivers, compared to the generalized motor problems, weakness and fatigue, and autonomy loss they have to cope with [55,56,69,70]. Finally, participants experiencing acute neurologic pathologies may perceive OD as a temporary symptom, requiring immediate clinical management rather than implying long consequences. Further studies are needed to more correctly evaluate the importance of OD, net of other symptoms, for patients and caregivers in different clinical conditions.

Despite evidence of increased physical and emotional burden levels among informal caregivers of elderly patients with OD [20], only few of the included studies investigated informal caregivers’ needs, a topic deserving further exploration.

Overall, the pursuit of the research aim–identifying perceived care needs–led to the retrieval of a very limited number of studies, reflecting a substantial lack of attention to patients’ and caregivers’ perspectives in both research and clinical domains. Nevertheless, as expected, a broad variety of OD-related needs and strategies for their satisfaction were identified by both patients and caregivers. Informational, social, and psychological needs were equally cited by patients and caregivers, whereas physical needs were reported only by patients and practical needs concerning strategies to manage OD were primarily quoted by caregivers.

Traditionally, OD research has been prominently focused on swallowing physiology, OD clinical assessment and management field [71]. Since the introduction of the ICF, more attention was paid to the manifold implications of OD [9–11], through scales investigating the psychological and social dimensions, patients’ perception of OD physical symptoms, and HRQOL [72,73]. Despite the usefulness of these quantitative measures towards a multidimensional standardized OD assessment, they do not allow for evaluating the importance attributed by individual patients to each OD related impairment or problem. Considering that need perception arises from the gap between the perceived severity of functional loss and the value attributed to the lost function [26], qualitative research instruments such as interviews and focus groups appear as the most appropriate instruments to investigate OD-related needs; related findings may subsequently guide the development of questionnaires specifically targeting those needs.

To the best of our knowledge, this is the first scoping review aimed at summarizing the available evidence on patients’ and caregivers’ OD-related needs, from a person-centered perspective. At the conceptual and interpretive levels, the lack of a solid, unifying and person-centered theoretical model guiding research hypotheses and result interpretation emerged from the analysis of the reviewed studies. In the present review, the Supportive Care Framework (SCF) [27], previously used for classifying needs reported by patients diagnosed with stroke and ALS [33,34], proved to be a satisfactory theoretical framework to categorize OD-related needs. In order to fit the results of the present review, only slight changes were brought to the framework. In particular, emotional and psychological needs were merged into the category “psychological needs” as, from a disciplinary perspective, emotions belong to psychological domain, together with cognition and motivational processes. In addition, in order to better inform future research and clinical practice, the SCF categories were articulated into OD specific sub-categories.

Based on the review findings, we also suggest the potential of the Common-Sense Model of Self-Regulation (CSM) [35,36] as a suitable conceptual and interpretive framework to guide research on patients’ and caregivers’ needs. The broad range of care needs emerged from the present review can be fruitfully interpreted through the CSM. Informational needs can be related to illness or treatment representations. For example, patients’ and caregivers’ difficulties in building a mental representation of OD based on their own experience, in terms of illness label, associated symptoms and timeline may elicit the need for more detailed, clearer or tailored information about these issues. Similarly, in order to form an adequate mental representation of treatment consequences, patients and caregivers may need more detailed information about PEG advantages. Furthermore, informational needs may guide action, as highlighted by the patients’ and caregivers’ need for more detailed and tailored information on OD-adapted recipes and OD management during holidays.

Practical needs may instead emerge from limitations in individual and environmental resources, such as restricted availability of safe and tasty food, or lack of competences in preparing meals.

Physical needs, such as maintaining body integrity or reducing pain while eating, may be linked to treatment representation (PEG and radiotherapy, respectively) and related consequences. The need for nutritionally adequate meals or proper oral hygiene may relate to the representation of OD causes (undernutrition could worsen OD through decreased muscles strength) and controllability (undernutrition could accelerate OD worsening in chronic conditions and slow recovery in acute ones).

Social needs may be primarily related to the representation of OD consequences. For instance, the changed meaning of food [12] may give rise to the need for social support and for changes in family and social roles. The physical and emotional caregivers’ burden consequent to OD [18,20] may lead patients to worry about the wellbeing of their caregiver.

Finally, psychological needs may be related to different components of the CSM. The need to cope with negative emotions and feelings of loss—frequently reported by persons with OD [9]—may be associated with OD emotional representation. Problem-focused strategies acted by OD patients may be linked to the CSM action plans aimed at best controlling OD symptoms and consequences. The need to accept a “new normal”, to maintain a positive attitude and to cultivate hope may be related to the representation of illness coherence.

Spiritual needs (i.e. “needs related to the meaning and purpose in life to practice religious beliefs” [27]), though not retrieved in the present review, could also be included in the CSM component of illness coherence. Studies involving a wider range of countries and cultural groups could contribute to a more comprehensive understanding of the potential role of spiritual needs in OD patients and informal caregivers.

Besides shedding light on patients’ and caregivers’ illness and treatment representations, the CSM could be a suitable framework at the intervention level. By taking into account the reciprocal interplay between mental representations and action plans, the former contributing to shape the latter and vice-versa, researchers and clinicians could design needs-driven interventions [59] based on the global representation of illness and treatment subjectively built by patients and caregivers.

Supporting information

Acknowledgments

The authors would like to acknowledge the speech and language pathologist, Dr Thais Alves, for the valuable contribution in the initial development of the search strategies and the charting data form.