Oral Care Considerations for People with Cystic Fibrosis: a cross-sectional study

doi:10.21203/rs.3.rs-1990331/v1

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Oral Care Considerations for People with Cystic Fibrosis: a cross-sectional study

https://doi.org/10.21203/rs.3.rs-1990331/v1

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Background: Cystic Fibrosis is the most common autosomal recessive genetic condition in the Caucasian population. Oral health in adults with Cystic Fibrosis is an area lacking in research, due to their historic low life expectancy. The age profile of people with Cystic Fibrosis (CF) is increasing, due in part to improved therapies. Further study is needed in order to ascertain their dental needs and improve provision of care to these medically vulnerable individuals.

Methods: A cross sectional survey was carried out. A structured, anonymous online questionnaire, with both quantitative and qualitative aspects, was used to obtain information regarding their feelings towards dentists and dental appointments. The final version of the questionnaire was based on a collaborative effort between researchers at Cork University Dental School and Hospital and Cystic Fibrosis patient advocates from Cystic Fibrosis Ireland. The inclusion criteria were people over the age of 18 years with a diagnosis of CF. Participants were recruited via CF Ireland’s mailing list and social media channels. The quantitative portion of the questionnaire underwent statistical analysis using IBM SPSS. The qualitative portion underwent inductive thematic analysis.

Results: A total of 71 adults with CF responded to the survey. 54.9% of respondents were unhappy with their teeth. 63.4% felt that CF had an impact on oral health. 33.8% were anxious about attending their dentist. 54.9% felt that dentists should be part of the multidisciplinary team. Respondents believed that CF has impacted on their oral health due to the medications and dietary requirements involved, as well as tiredness and other side effects of CF. Some reasons for being anxious about attending the dentist involve cross infection concerns, issues with the dentist, with tolerating treatment, and with the teeth themselves. They wanted dentists to be aware of the practicalities of dental treatment for people with CF, especially their discomfort with lying back. They also want the dentist to be aware of the impact that their medication, treatment and diet has on their oral health.

Conclusions: Over a third of PWCF reported anxiety about attending the dentist. Reasons for this included fear, embarrassment, cross infection concerns and problems with treatment, especially being in the supine position. PWCF want dentists to be aware of the impact that CF can have upon dental treatment and oral health care.

Cystic Fibrosis

CFTR modulators

Oral Health

Dentistry

Cystic Fibrosis (CF) is an autosomal recessive genetic condition, resulting from mutations in the cystic fibrosis transmembrane conductance regulator (CFTR) gene on chromosome 7 [1] Its dysfunction results in abnormal transport of Chloride and Bicarbonate ions, leading to thick viscous secretions in multiple organ systems. This can result in diabetes, osteoporosis, liver disease, chronic kidney disease, and gastro-intestinal issues, as well as serious respiratory problems, with progressive bronchiectasis and recurrent infective pulmonary exacerbations being the hallmark of CF lung disease [2]. Lung transplantation is recommended for individuals with advanced lung disease [3]. Due to earlier diagnosis and improved therapies, the life expectancy of people with CF (PWCF) has greatly improved, from a median survival age of 11 years in 1978 to between 44 to 53 years in 2019 [4]. Due to the fact that CF has changed from an almost exclusively paediatric disease to a disease of adulthood, the transition to adult care is now an important aspect of CF patient management [5]. The medical care of PWCF is complex, with treatment aiming to control the symptoms, reduce complications and reduce the severity of different manifestations of CF. Oral care considerations include the long-term use of antibiotics (which may be inhaled or nebulised), bisphosphonates for osteoporosis, presence of CF related diabetes, gastro-oesophageal reflux disorder, and malnutrition which may be treated with Oral Nutritional Supplements with a high sugar content [6–10].

The oral health status of individuals with CF is not yet fully understood. A 2020 systematic review showed that the majority of studies show better oral hygiene, with lower levels of gingivitis and plaque among PWCF than controls. [11], with fewer studies showing increased gingivitis and higher levels of plaque and calculus. The vast majority of research was carried out on children [11]. Studies have shown that people with special medical care needs may be less likely to access dental care [12] and a recent report has shown that people with Cystic Fibrosis may attend dental practices less frequently than is recommended [13].

Aims

This study seeks to investigate the attitudes of PWCF towards dental attendance and any perceived barriers to treatment.

Study Design

A cross sectional survey was carried out in accordance with the World Medical Association Declaration of Helsinki and received Ethical Approval from the Clinical Research Ethics Committee of the Cork Teaching Hospitals (ECM 03/2022 PUB).

The inclusion criteria were people over the age of 18 years with a diagnosis of CF. The exclusion criteria were people without Cystic Fibrosis, or people with Cystic Fibrosis under the age of 18. All participants gave written consent to take part, declaring that they understood they were under no obligation to complete the questionnaire and that they consented to data collection. Participants were recruited via Cystic Fibrosis Ireland’s mailing list and social media channels.

Questionnaire

A structured, anonymous online questionnaire, with both quantitative and qualitative aspects, was used to obtain information regarding their feelings towards dentists and dental appointments. Patient involvement was sought from patient advocates from CF Ireland, with whom the questionnaire was trialed and amendments made based on their feedback. The final version of the questionnaire was based on a collaborative effort between researchers at Cork University Dental School and Hospital and CF patient advocates from CF Ireland. Data was collected from May to August 2020. A copy of the questionnaireis included in Appendix I.

Data analysis

Descriptive statistical analysis of the quantitative portion was completed using IBM SPSS (v26; SPSS Inc., Chicago, IL, USA). The qualitative portion, which consisted of open-ended questions, underwent thematic analysis as described by Braun and Clarke [14] (2006). The analysis was inductive in that there is no previous study in this area and, therefore, the data collected determined the themes generated. Selected quotations and related themes can be seen in Appendices II-IV.

A total of 71 adults with CF responded to the survey.

The quantitative responses are summarised in Table 1.

Table 1

Respondent Profile
Variable	Category	N	%
Gender	Male Female	33 38	46.5 53.5
Level of education completed	Primary Level Secondary Level Third Level	3 17 48	4.2 23.9 67.6
Employment	Full-/part- time employment/self-employed Unemployed Prefer not to say	43 25 3	60.6 35.2 4.2
Natural teeth	20 or more 10–19 Don’t know	64 4 3	90.1 5.6 4.2
Denture	Wears denture No Denture	0 71	0 100
Are you happy regarding the appearance of your teeth?	Yes No Declined to answer	30 39 2	41.3 54.9 2.8
Dental attendance	Regular attender Irregular attender	42 29	59.1 40.8
Reason for last dental visit	Routine check-up/consultation Pain/trouble with teeth gums or mouth Treatment	39 21 8	54.9 29.6 11.3
Do you think that CF has impacted your oral health?	Yes No Don’t know/Declined to answer	45 13 13	63.4 18.3 18.3
Anxious/worried about attending dentist?	Yes No Don’t Know	24 45 2	33.8 63.4 2.8
Do you make your dental practice aware of your CF status?	Yes No Dental practice already aware	25 19 27	35.2 26.8 38

The majority of respondents had a high level of education (67.6% having 3rd level education) and were employed or self-employed (60.6%). They retained the majority of their teeth and no respondent had a denture. Despite this, the majority of patients (54.9%) were unhappy with the appearance of their teeth. 40.8% of people were irregular attenders and nearly 30% said the reason for their last dental visit was “pain or trouble with teeth, gums or mouth”. The majority (63.4%) of patients felt that CF had an impact on their oral health. 33.8% were anxious about attending the dentist. 25.2% of respondents made the dental practice aware of their CF status before attending, with a further 38% reporting that the dental practice was already aware.

When asked if they think dentists should be part of the multidisciplinary team, 54.9% said “Yes”, 32.4% said “Maybe” and only 12.7% said “No”.

Qualitative data

Three questions underwent thematic analysis and will be dealt with separately. These were:

1. Do you believe that CF has impacted on your oral health in any way?

2. Are you anxious/worried about going to the dentist? If yes, why?

3. What do you think is important for the dentist to know about CF?

Question 1: Do you believe that CF has impacted on your oral health in any way?

The majority of respondents (63.4%) believed that CF did have an impact on their oral health. The major themes that emerged from this question, in decreasing frequency was:

Impact of medications (especially antibiotics)
Tiredness
Diet
Side effects of CF

The main CF-related factor that respondents mentioned was the impact of medications- concerns ranged from nausea/vomiting to discolouration, to dry mouth. Many blamed antibiotics in particular as the cause for “staining”, “lost enamel” and “weakened teeth”.

Tiredness/lack of energy also had an impact on their oral health. Many mentioned being “too tired to brush”, “no energy to brush”, with one respondent noting “When I've been very sick it's hard to get the energy to brush your teeth, I remember having to ask my mum to brush them for me”.

Another area where CF impacted oral health was diet. Many respondents noted an increase in amount of sugar needed: “to get calories in”, “just over eating sweet things when sick and no real appetite for large meals”, and being “addicted to sugar”. One respondent noted that they had an increased need for sugary food since developing CF related diabetes (CFRD) “Since developing CFRD, I eat a lot more jellies and drink orange juice now to treat lows [hypoglycaemic episodes].

Other systemic effects of CF also had an impact on their dental health, such as “tummy issues causing bad breath and erosion” and missing dental appointments due to being unwell.

Question 2: Why are you anxious/worried about going to the dentist?

Five major themes emerged from this question, in descending order of frequency, these were:

Issues with the dentist
Cross-infection concerns
Problems with treatment
Problems with teeth
CF related problems.

Dentist-related problems included fear of dentists themselves, fear of being judged by the dentist, with some respondents noting “I have previously been treated with lack of understanding from dentists/hygienists” and ““I am always in trouble when I attend”.

Another aspect of dental visits that concerned PWCF was cross-infection control, with many respondents mentioned increased anxiety since the Covid-19 pandemic “I did not feel this way before but I do now in light of Covid-19”. Some respondents also mentioned concern at being so “close to the sink at dentist chair”. This is due to the perception that there may be stagnant water or saliva in the sink [spittoon] which could pose a cross-infection risk to the individual.

A practical issue that caused anxiety in PWCF was dental treatment, with many expressing difficulty in lying in the supine position that is usually employed during a dental examination- “I find it difficult to be in a flat position as it affects my breathing. It's also very difficult not to cough”. Some respondents reported dental problems causing anxiety surrounding dental appointments: “My teeth are all wearing away at the bottom the past few years and I feel my teeth are going to break soon”, “I just feel embarrassed about my teeth”.

CF itself had an impact on people’s anxiety regarding dental attendance, with one individual noting that they “Feel sick enough without there being another thing wrong with my teeth”.

Question 3: What do you think is important for the dentist to know about CF?

4 major themes emerged from this question. In descending order of frequency, they were:

Practicalities of dental treatment
Impact of medication/treatment/diet
Impact of CF itself,
Cleanliness of surgery/cross-infection control.

The main concerns PWCF had were the practicalities of dental treatment, namely keeping the patient upright and allowing them to have breaks during treatment. The vast majority of respondents said some variant of this, e.g., “it is often difficult for people with CF to lie down and not cough for long periods of time”, “would have liked option to sit more upright during prolonged treatment, felt this was where I was exposed to risk of aspiration”, “I can't lie flat or sometimes breathe through my nose quick enough”. On a positive note, some respondents noted that their dentist/hygienist already accommodates this: “My dentist is very good and tilts chair upright and also pauses when I indicate I need to cough. It slows process but he is very understanding and allows time”, “Extra breaks may be needed, my dentist/hygienist are good when it come to that!”.

Another issue that PWCF wanted dentists to be aware of is the impact that medication, treatment and diet can have on their oral health. A concern is that dentists are not aware or not understanding of the effects that CF-related medication and diet has on their oral health. They want dentists to be aware of “the full extent of the treatment required that may impact the teeth, gums and tongue”, “the effect different meds have on oral hygiene”, “before lecture on oral hygiene, sweets etc.- CF patients need [them] to maintain weight”. Another overlapping theme that emerged was for the dentists to be aware of CF itself and the impact that has on the individual: “the impact it [CF] has on our health and mental health and how that could relate to our dental hygiene/care/condition”, “About CF in general and how CF has impacted our oral health”.

The final theme that emerged and can be linked to dental anxiety as outlined in the previous section, is for dentists to be aware that PWCF may be anxious about cross-infection risks. Respondents mentioned aspects such as “cross-infection risks, especially infection risk from pseudomonas”, “clean lines”, “make sure all equipment is sterile for treatment”.

PWCF can experience many barriers to accessing dental care which may result in irregular attendance. The most common barriers described by participants of this study include dental anxiety, cross-infection concerns, and difficulty with the practicalities of treatment, for example, laying back in the dental chair.

33.8% of respondents said they were anxious regarding attending the dentist. It is a much higher proportion than the estimated 4–20% of people in the general population in developed countries who suffer from dental anxiety [15, 16]. It is also a higher proportion than a 1998 sample of people with special health care needs where 27.9% reported “fear/anxiety about dental visits” Ideally, levels of dental anxiety amongst the population would be decreasing year on year due to new therapies etc, therefore, this is a major concern. It is well established that dental anxiety is one of the most important barriers to dental care [17, 18]. Therefore, this study is important to understand what is causing the high level of anxiety in PWCF.

Examining the major themes regarding this, it can be seen there are dental-related concerns (the dentist themselves and treatment concerns) and patient-related concerns (problems with teeth and CF as a condition). Clinicians should seek to rectify the concerns over which they have control, i.e. being judgmental regarding patient’s teeth or need to stop for breaks.

This study shows that PWCF themselves can be concerned regarding the appearance of their teeth, and many believe the medication and diet related to CF has had a negative effect on their oral health. They have raised concerns about dentists’ lack of understanding on the condition and its management, and therefore, may be anxious about attending a dentist due to fear of being adversely judged. PWCF want dentists to understand that due to increased dietary needs, periods of ill-health and polypharmacy, they may have increased risk of oral disease.

One of the major points repeated was the need to sit the patient in an upright position due to a frequent need to cough, this is due to mucus build up or reflux, and is reported by almost every person with CF [19, 20]. Therefore, sitting the patient in an upright position during treatment should be accommodated if possible.

Dentists should also alleviate the patient’s concern regarding cross-infection control, by reassuring PWCF that the strictest standards of cross-infection control are being adhered to.

A large number of respondents mentioned Covid-19 specifically as an area of concern, this is in-keeping with recent studies which showed that even in people without CF, “those with high COVID-19 fear were at least six times more likely to not visit the dentist” [21].

This study is important as it provides the first insight into the attitudes of PWCF towards dental treatment and gives recommendations for improving provision of care to these patients. There is a lot of overlap between what PWCF want dentists to know, and what causes dental anxiety in them. Therefore, if dental practitioners are educated about CF and its repercussions, and make small amendments to their practice based on these, it should make the dental experience less anxiety-inducing for PWCF.

Strengths and limitations

A strength of this study is that it is anonymous, therefore respondents were more likely to be honest with how they perceived the dentist and dental appointments. The fact that we employed the use of PPI (Patient and Public involvement) was a major benefit as a 2014 systematic review shows PPI has a a positive impact on all research/stages [22].

A limitation is the relatively small sample size. It should be noted there are 746 adults with CF in Ireland as of 2019 [23], a response from approximately 9.5% of that population was obtained.

A third of PWCF reported anxiety about attending the dentist. Reasons for this included fear, embarrassment, cross-infection concerns and problems with treatment, especially being in the supine position. PWCF want dentists to be aware of the impact that CF can have upon dental treatment and oral health care.

Recommendations

Medical complexities faced by people with CF such as frequent and long-term antibiotic use, specific dietary requirements and periods of illness may lead to difficulty in maintaining meticulous oral hygiene. Dentists should be empathetic and should endeavour to make the patient experience more comfortable by sitting the patient upright if possible to facilitate mucus clearance.

CF- Cystic Fibrosis

CFTR- cystic fibrosis transmembrane conductance regulator

PPI- Patient and Public Involvement

PWCF- Person/People with Cystic Fibrosis

Ethics approval and consent to participate

This study carried out in accordance with the World Medical Association Declaration of Helsinki and received Ethical Approval from the Clinical Research Ethics Committee of the Cork Teaching Hospitals (ECM 03/2022 PUB). All participants gave informed written consent to take part.

Consent for publication

Not applicable- data is anonymised

Availability of data and materials

The datasets generated during and/or analysed during the current study are not publicly available due to patient confidentiality but are available from the corresponding author on reasonable request.

Competing interests

The authors declare that they have no competing interests

Funding

This study received funding from Cystic Fibrosis Ireland, Health Research Board Funding (CSF-2020-003) and CiSA funding from University College Cork.

Authors' contributions

NC was responsible for the concept, study design, analysis and creation of the manuscript.

FOL was involved in the concept, study design and acquisition of data. AR was involved in the supervision of the manuscript draft and revisions. FB was involved in the supervision of the manuscript draft and revisions. BP was involved in the concept and acquisition of data. MH was involved in the concept, study design, acquisition of data, manuscript draft and revisions.

All authors approved the submitted version.

All authors agreed both to be personally accountable for the author's own contributions and to ensure that questions related to the accuracy or integrity of any part of the work, even ones in which the author was not personally involved, are appropriately investigated, resolved, and the resolution documented in the literature.

Acknowledgements

The authors wish to acknowledge the support given by CF Ireland, both through their patient advocates in the creation of the questionnaire, and their distribution of the questionnaire via their social media channels and mailing list.

Merjaneh L, Hasan S, Kasim N, Ode KL: The role of modulators in cystic fibrosis related diabetes. Journal of Clinical & Translational Endocrinology 2022, 27:100286.
Ronan NJ, Elborn JS, Plant BJ: Current and emerging comorbidities in cystic fibrosis. La Presse Médicale 2017, 46(6):e125-e138.
Ramos KJ, Smith PJ, McKone EF, Pilewski JM, Lucy A, Hempstead SE, Tallarico E, Faro A, Rosenbluth DB, Gray AL et al: Lung transplant referral for individuals with cystic fibrosis: Cystic Fibrosis Foundation consensus guidelines. Journal of Cystic Fibrosis 2019, 18(3):321–333.
McBennett KA, Davis PB, Konstan MW: Increasing life expectancy in cystic fibrosis: Advances and challenges. Pediatr Pulmonol 2022, 57 Suppl 1(Suppl 1):S5-s12.
Scotet V, L'Hostis C, Férec C: The Changing Epidemiology of Cystic Fibrosis: Incidence, Survival and Impact of the CFTR Gene Discovery. Genes (Basel) 2020, 11(6).
Taccetti G, Francalanci M, Pizzamiglio G, Messore B, Carnovale V, Cimino G, Cipolli M: Cystic fibrosis: Recent insights into inhaled antibiotic treatment and future perspectives. Antibiotics 2021, 10(3):338.
Marquette M, Haworth CS: Bone health and disease in cystic fibrosis. Paediatric Respiratory Reviews 2016, 20:2–5.
Robinson NB, DiMango E: Prevalence of Gastroesophageal Reflux in Cystic Fibrosis and Implications for Lung Disease. Annals of the American Thoracic Society 2014, 11(6):964–968.
Bridges N: Diabetes in cystic fibrosis. Paediatric respiratory reviews 2013, 14:16–18.
Victoria C-B, Casilda O, Nuria P, José A-F, María G-O, José S-TF, Victoria GM, Ignacio R-G, Gabriel O: Oral Nutritional Supplements in Adults with Cystic Fibrosis: Effects on Intake, Levels of Fat-Soluble Vitamins, and Bone Remodeling Biomarkers. Nutrients 2021, 13(2).
Coffey N, O’ Leary F, Burke F, Roberts A, Hayes M: Periodontal and Oral Health Status of People with Cystic Fibrosis: A Systematic Review. Journal of Dentistry 2020:103509.
Alfaraj A, Halawany HS, Al-Hinai MT, Al-Badr AH, Alalshaikh M, Al-Khalifa KS: Barriers to Dental Care in Individuals with Special Healthcare Needs in Qatif, Saudi Arabia: A Caregiver's Perspective. Patient Prefer Adherence 2021, 15:69–76.
Coffey N, O' Leary F, Burke F, Plant B, Roberts A, Hayes M: Self-reported Dental Attendance, Oral Hygiene habits and Dietary Habits of Adults with Cystic Fibrosis in the Republic of Ireland. Special Care in Dentistry 2022.
Braun V, Clarke V: Using thematic analysis in psychology. Qualitative Research in Psychology 2006, 3(2):77–101.
Locker D, Thomson W, Poulton R: Psychological disorder, conditioning experiences, and the onset of dental anxiety in early adulthood. Journal of dental research 2001, 80(6):1588–1592.
Sukumaran I, Taylor S, Thomson WM: The prevalence and impact of dental anxiety among adult New Zealanders. International Dental Journal 2021, 71(2):122–126.
Freeman R: Barriers to accessing dental care: patient factor. British Dental Journal 1999, 187(3):141–144.
Hill KB, Chadwick B, Freeman R, O'Sullivan I, Murray JJ: Adult Dental Health Survey 2009: relationships between dental attendance patterns, oral health behaviour and the current barriers to dental care. British Dental Journal 2013, 214(1):25–32.
Fathi H, Moon T, Donaldson J, Jackson W, Sedman P, Morice AH: Cough in adult cystic fibrosis: diagnosis and response to fundoplication. Cough 2009, 5:1–1.
Penketh A, Wise A, Mearns M, Hodson M, Batten J: Cystic fibrosis in adolescents and adults. Thorax 1987, 42(7):526–532.
González-Olmo MJ, Delgado-Ramos B, Ortega-Martínez AR, Romero-Maroto M, Carrillo-Díaz M: Fear of COVID-19 in Madrid. Will patients avoid dental care? Int Dent J 2022, 72(1):76–82.
Brett J, Staniszewska S, Mockford C, Herron-Marx S, Hughes J, Tysall C, Suleman R: A systematic review of the impact of patient and public involvement on service users, researchers and communities. Patient 2014, 7(4):387–395.
CF Annual Report 2018 [https://www.cfri.ie/docs/annual_reports/CFRI2018.pdf]

No competing interests reported.

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Oral Care Considerations for People with Cystic Fibrosis: a cross-sectional study

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Abstract

Introduction

Aims

Methodology

Study Design

Questionnaire

Data analysis

Results

Qualitative data

Discussion

Strengths and limitations

Conclusions

Recommendations

List of abbreviations

Declarations

References

Additional Declarations

Supplementary Files

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