Oral submucous fibrosis is an oral potentially malignant disorder1 characterized by burning sensation in the oral cavity, oral ulceration, vesiculation, blanching, and stiffening of the oral mucosa and oropharynx, consequently leading to gradual limitation of mouth opening. Shevale et al2 reported increasing number of OSF cases every year, affecting 5 million people in India alone (0.5% of the Indian population). Another study conducted by Hazarey et al3 in the central part of India recorded 21.9 OSF cases per 1,000 individuals with male predominance at the ratio of 4.9:1. Several factors contribute to this increasing prevalence of OSF, especially in India, including addiction of youth population to this socially acceptable and popular habit of areca nut and its preparations, easy access, effective price changes, and marketing strategies.4,5 Mortality rate is significant because it transforms into oral cancer, particularly squamous cell carcinoma at a rate of 7 to 30%.2
Quality of life is a vital and often-required health outcome measure that is relevant to the patients care. The QoL is a general term integrating several aspects of life, such as physical, psychological, social, economic, spiritual, cognitional, and sexual dimensions. A disturbance in any one aspect will in turn affect the other domains and this influences the overall QoL.6 The QoL assessment should be part of the evaluation of oral health in day-to-day practice.7 Oral submucous fibrosis patients experience significant health-related symptoms including reduced mouth opening, burning sensation, and inability to eat, which are quite unique as compared with other oral pathologies. Along with social and emotional distress, these patients are always afraid of developing a cancer resulting in increased psychiatric morbidity.8 Thus, OSF definitely has an impact on QoL of affected individuals. Because of the uniqueness of OSF in terms of clinical presentation as well as subjective symptoms, it warrants special requirement for QoL assessment.
There is paucity of literature on QoL assessment in OSF patents9 in spite of its higher prevalence2,3 and malignant potential.2 This might be due to unavailability of a condition-specific QoL measure for OSF. Patient-reported outcome measures were identified as a research priority and have been routinely used nowadays. A PROM reflects a patient’s perceptions of general health and well-being; which are routinely quantified by administration of questionnaires.10 Past studies used generic questionnaires for the assessment of QoL in OSF patients.11-13 However, these generic instruments lack the sensitivity to evaluate the QoL accurately, as they are applicable to a wide variety of populations and disease states. The necessity of the work in this area has recently been recognized by Rai.14 With this dire need in mind, the present study was designed to focus on the outcomes and experiences of the OSF patients with a view to developing a proper PROM for OSF patients specifically.
MATERIALS AND METHODS
Institutional Ethics Committee approval was obtained before the commencement of the study and all the patients signed the written informed consent. A total of 30 clinically diagnosed OSF patients were recruited in the study irrespective of the age, gender, education, occupational status, income, and severity of OSF by purposive sampling method. The OSF cases were categorized based on the following clinical criteria: intolerance to hot and spicy foods, pale-looking oral mucosa, palpable fibrotic bands, and chronic progressive trismus.15,16 Inclusion criteria included patients above 18 years of age, who were willing to participate, able to understand, and complete the questionnaire on their own. Exclusion criteria for the study included patients with any systemic disorder, any oral lesion except OSF, and the presence of clinically suspicious malignant changes. A thorough case history was recorded including the sociodemographic information, and diagnosis of OSF was made by clinical examination. Interincisal mouth opening was measured using a digital Vernier caliper. For burning sensation, the visual analog scale score was recorded. The severity of OSF was graded based on the degree of mouth opening into stage I (35–40 mm), stage II (30–34 mm), stage III (20–29 mm), and stage IV (< 20 mm).17
A grounded theory approach18 with constant comparison was used throughout the research. Open-ended interviews and focus group discussions were performed with 30 adults with OSF to identify the factors that patients view as important for their oral health and overall well-being. These patients were characterized by a wide degree of disease severity and diverse range of sociodemographic profile. A single researcher performed 14 personal (one-to-one) interviews and four focus group discussions in the Marathi language (the native language of the study population) in nonclinical settings. Interviews were open, semi-structured in nature with prompts.19 Patients were motivated to talk freely about their oral health-related problems in focus group discussions.20 The prompts encouraged exploration of patients’ perceptions on the history, diagnosis, and progression of OSF. Interview prompts (Table 1) were informed by the clinical experts and patient input. Relevant topics were pooled from thorough literature review. Additionally, a panel of eight experts (specialist in oral medicine, oral pathology, oral surgery, and psychology) was approached using the Delphi technique21 to understand the affected aspects of patient’s life due to OSF. The duration of each interview was around 20 minutes and each focus group discussion lasted for more than half an hour. None of the recruited patient had any preexisting relationship with the interviewer in any context. Interviews and focus group discussions were audio recorded. The field notes were recorded to document particular details about the process. The recording was then anonymized, transcribed, and translated in English at the same time.22 A saturation table was prepared using the data from each patient (Table 2).
Data analysis was performed by using an in-depth narrative thematic analysis method.23 Data analysis instituted prior to completing all the interviews to control topic saturation. The data saturation occurred in 10th interview. The data were coded initially and then categorized into themes and subthemes. Initial coding of the interview transcripts was performed independently by two researchers to increase the reliability of data analysis.24 Themes evolved from recurring words and ideas from the patients.25 This part of the analysis was performed by two independent researchers. Intercoder reliability was calculated for each theme and overall coding using percentage agreement and the Kappa statistic. Themes were discussed and granted by agreement of all the authors. The themes were then discussed in the interdisciplinary research team to achieve intersubjective validity of the results.
Demographics and Clinical Outcome
The age of the participants ranged from 20 to 65 years with mean of 33.50 years. Males were more prevalent than females with a male-to-female ratio of 9:1. Mouth opening ranged from 2 to 37 mm with mean of 19.7 mm. Majority of the participants were graded as stage III (53.33 %) followed by stage II (23.33%), stage IV (16.66%), and stage I (6.66%). Only few (20%) of the study participants were graduates (having bachelor’s degree) or had diplomas. A great number (80%) of the participants possessed lower routine and semi-routine occupations and more than half (66.66%) belonged to lower income category (Table 3).
Patient-reported Outcome Measure Analysis
Four main themes emerged from the data analysis: (1) discomfort and functional impairment; (2) psychological wellness; (3) physical wellness, and (4) social wellness. Kappa agreement for all the themes ranged from 0.65 (social wellness) to 0.72 (discomfort and functional impairment) with significant overall kappa agreement (0.67) for all the themes together (Table 4).
Discomfort and Functional Impairment
This theme was the key area of importance for most of the respondents. Two subthemes evolved: discomfort and functional impairment.
The OSF patients had come across several troubles in view of discomfort and functional impairment. Difficulty in mouth opening was the most common complaint reported by all the participants. The diverse amount of mouth opening was reported as per the degree of severity of the disease. A 19-year-old nervous male OSF participant discussed, “I am not able to open my mouth completely as like my friends. I face difficulties during brushing my teeth, especially posterior ones because of reduced mouth opening.” Another participant (36-year-old female) reported “being unable to eat big bites of foods due to limited mouth opening, I think, I am undernourished as I have to be dependent on liquid diet most of the times.”
The second most common complaint reported was burning sensation. Majority of the participants experienced burning sensation in the oral cavity. “I am experiencing very severe burning sensation; I used to enjoy my favorite foods (naming some Indian spicy dishes) which I do not anymore now. Everyone in my family have to eat less spicy foods as same food is being cooked for everyone in my family” (29-year-old male). One young male participant (20-year-old) with stage IV disease informed, “Being foodie person, I used to visit restaurants and street food courts quite frequently. Recently, my burning sensation has increased to such an extent that I have stopped visiting such places.”
Few patients reported changes in their taste sensation. A 23-year-old female participant discussed “I think, I am feeling a bit of dryness in my mouth since few months and find difficulties in recognizing a taste of food sometimes.” Another male participant 32 years of age expressed, “I want to get rid of this my mouth condition, it started with burning sensation and my taste sensation has also altered these days. I am foodie person and I do not enjoy my dearest food dishes since last 2 years.” Its very disheartened to listen when one participant described his worsened taste as “I am eating just for the sake of living” (50-year-old male).
I: Interview; FG: Focus group; X: Interview in which a new concept was discussed
SD: Standard deviation
|Discomfort and functional impairment||0.72||99.42||<0.001|
Three (10%) participants expressed concern at being unable to speak clearly. A 49-year-old male participant explained, “I feel along with my reduced mouth opening, its getting difficult for me to pronounce some words clearly. My dentist informed me regarding a bit of immobility of my tongue when this my mouth condition got diagnosed for the first time.” Another male participant (29-year-old) discussed “my family and few friends noticed unclear words when I talk to them.” Intriguingly, a 34-year-old male sales representative with a stage IV disease reported, “I am not able to convince my clients efficiently due to troubles with pronunciation and clarity of words, which has significantly affected my business.”
Many of the participants (76%) experienced psychological imbalance because of their mouth condition. Initially, some patients were shocked to discover their mouth condition. Most of them revealed their fear after knowing about the severity of the disease.
“I am worried and feel frustrated because of reduced mouth opening, burning sensation and difficulty in eating food. As soon as I diagnosed with the OSF and come to know its potential of turning into cancer, I am continuously thinking about the chances of having cancer in my mouth. Since that day, I am looking for different ways to cure the condition leading to difficulty in concentrating on my routine work and loss of enthusiasm in life” (35-year-old male).
Another 46-year-old participant described, “because of reduced mouth opening, it is difficult for me to brush all teeth and maintain good oral hygiene. The bad odor from my mouth frustrates me and creates embarrassing situations many times at my work place as well as at home. This also results in feeling of low esteem and creating loss of interest and loneliness these days.”
One young 21-year-old college-going student admitted that “the stress of studies is one of the reason for starting kharra/gutkha (popular areca nut preparations in India) chewing habit. After three years, I noticed limited mouth opening. As I learnt about this condition over the internet, it aggravated my stress as I got scared of developing a cancer. However, after accepting the fact, I was relieved and looking forward to life positively with treatment of my mouth condition.”
A 27-year-old unmarried female participant expressed, “I feel depressed and rejected due the reduced mouth opening. Lack of appetite and reduced food intake because of burning sensation and loss of taste results in weight loss. My parents always express concern regarding delays in my marriage. We tried multiple doctors for speedy recovery of the condition and to avoid further complications, such as cancer.”
Four (13.33%) male participants reported changes in their sleep patterns. A 24-year-old male participant explained, “It was a horrible night for me when I come to know about this mouth condition from my dentist. I was awake almost all hours of that night.” Another participant (29-year-old male) expressed, “in the initial days after diagnosis I could easily sleep at night, but as this condition causing newer consequences day-by-day, its been disturbing my sleep these days.” Remaining two participants were also reported of being out of their sleep routines. Other participants explained of having decent, comfortable night’s sleep.
Only 2 (6.66%) male participants reported changes in their level of fatigue and need for more rest. Both of them explained as “feeling overall weakness due to mouth condition. Very limited mouth opening and burning sensation in the mouth restrains from eating food and feel like to have more rest during daily routine work.”
This is another important emerged theme. Majority of the participants (76%) deal off with their compromised social interactions.
A 25-year-old male participant expressed “though I love to join friends and relatives frequently, I make an effort these days to stay away from parties and functions. I feel offended as I am unable to eat normal food like others due to my restricted mouth opening and burning sensation.” Another participant (42-year-old) described “it is very humiliating for me to ask for less spicy food to my wife in front of my children. Also, it is very embarrassing for me when my parents ask about the reasons for not being able to open the mouth completely. I feel like I am getting somewhat away from my family members.”
We noticed that majority of the participants started habit of kharra/gutkha chewing due to peer pressure. However, very few participants pointed out enjoyment factor as the reason for starting the habit. A great number of participants felt “sense of well-being” or stimulating effect immediately after kharra/gutkha chewing. Participants reported mixed feelings regarding experiencing stress relief after kharra/gutkha chewing. Unfortunately, many participants expressed “I feel agitated when cannot chew kharra/gutkha if needed. I quit this habit many times but unable to overcome the craving.”
The World Health Organization (WHO)26 states, “Health is a state of complete physical, mental, and social wellbeing and not merely the absence of disease and infirmity.” Oral health-related QoL (OHRQoL) is an integral part of general health and well-being and is recognized by the WHO as an important segment of the Global Oral Health Program. Past studies reported a negative impact of OSF on OHRQoL of many patients.8,11-13,27 and still further research is needed in this area, especially in South Asian countries, where prevalence of habit-related OSF is high. Unfortunately, at present, such attempts are restrained by an unavailability of a proper validated condition-specific PROM for OSF. Recently, Tadakmadla et al28 developed oral potentially malignant disorders QoL (OPMDQoL) questionnaire and found it valid and reliable to assess QoL in OPMDs like oral lichen planus, oral leukoplakia, and OSF. However, these three oral mucosal lesions have varying signs and symptoms.14 Thus, there is a clear need of more research in this field to bring clarity and validity to the subjective symptoms experienced by OSF patients. Rai14 suggested that future researches should develop and validate a condition-specific OHRQoL measure for OSF.
The utilization of PROs is an emerging metric and is becoming an increasingly important in health care.29 A PROM is an instrument used to quantify health-related QoL outcome variables from patient’s perspectives.30 Thus, PRO is a patient-reported assessment of health status rather than observer-reported health-related QoL data.31 The OHRQoL is a PRO that reflects an integral aspect of general health and well-being.32 This study involves our first step toward the development of an OHRQoL-OSF, specifically for OSF.
This study incorporated both personal interviews and focus group discussion to collect qualitative data. Personal interviews help in comprehensively attaining information on an individual’s personal experience. However, focus groups facilitate patients to share their experiences by using other’s ideas as cues. We have used semi-structured interviews as they offer flexibility and are less time consuming. The data saturation was achieved in the 10th interview which confirms the adequacy of sample size. Also, patients with diverse range of characteristics, such as age, gender, education, employment status, occupation, income, and varying levels of disease severity were considered.
Four main themes were identified: discomfort and functional impairment, psychological wellness, physical wellness, and social wellness.
The most distressing problem experienced by the participants was limited mouth opening. Burning sensation was the second most common complaints of the patients. This study found patients dealing with burning sensation while having food in combination with altered or worsened taste sensation. These above-mentioned problems altogether made participants to change their eating habits and to avoid their food of choice. Few participants also experienced discomfort during talking in the form of unclear words. Psychological distress was noticed to be prevalent in our participants. Some participants got scared immediately after diagnosis of their mouth condition as they were aware about OSF and its consequences. Others when they came to know regarding malignant potential of OSF were always worried of possibility of having mouth cancer. The term “cancer” is itself associated with fear and stigma in India.33 Few patients reported lack of interest and enthusiasm in life while others felt low esteem and embarrassment at their workplaces. Sleep disruption has been recognized as detrimental by some participants. Problems with sleep as a direct result of fear of OSF and cancer have been explored in these participants. Many participants reported their mouth condition as having a debilitating effect on their social interaction. Tadakmadla et al34 noticed the greater impact of OPMDs on QoL with physical impairment and functional limitations as the most distressing complaints. They also reported affected psychological and social wellness of the patients.
Thus, the overall scenario demonstrated impact of OSF on different aspects of individual’s life. “Discomfort and functional impairment” was noticed to be most recognized theme by our participants. However, OSF also has impacts on other important domains, namely, psychological, social, and physical wellness of the participants. Looking at the reports of this study, patients’ perspectives should be taken into consideration along with thorough clinical examination to best describe the sense of well-being of an individual and to decide the overall health care needs of the patients. Some of the limitations of the study included that the demographic distribution of participants was very homogeneous due to the local patient population.
These interview themes and issues procured from the focus group discussions were used to develop potential candidate items for a new condition-specific questionnaire (OHRQoL-OSF) for OSF patients. Patient-reported issues can give vital insights into patient’s perspectives through their experiences. The OHRQoL-OSF questionnaire was subsequently tested, refined, and validated to use as a clinical assessment tool for OSF patients specifically. We presume that future studies can be conducted across various cultural and language contexts by administering this questionnaire.