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characteristics, including cancer phobia, self-efficacy, pain- .... temic causes for oral burning22 were founded by detailed clinical evaluation and laboratory tests.
Odontology (2010) 98:160–164 DOI 10.1007/s10266-010-0123-6

© The Society of The Nippon Dental University 2010

ORIGINAL ARTICLE Hirofumi Matsuoka · Mika Himachi · Hirokazu Furukawa Shiho Kobayashi · Harumi Shoki · Ryo Motoya Masato Saito · Yoshihiro Abiko · Yuji Sakano

Cognitive profile of patients with burning mouth syndrome in the Japanese population

Received: September 21, 2009 / Accepted: January 25, 2010

Abstract The present study investigated which cognitive characteristics, including cancer phobia, self-efficacy, painrelated catastrophizing, and anxiety sensitivity, affect burning mouth syndrome (BMS) symptoms in the Japanese population. A total of 46 BMS patients (44 women and 2 men; mean age, 59.98 ± 9.57 years; range, 30–79 years) completed a battery of questionnaires, including measures of pain severity, oral-related quality of life (QOL), stressresponse, pain-related catastrophizing, self-efficacy, anxiety sensitivity, and tongue cancer phobia. The Pain CatastrophH. Matsuoka Health Sciences University of Hokkaido Hospital, Sapporo, Hokkaido, Japan M. Himachi Faculty of Human Cultures and Sciences, Fukuyama University, Fukuyama, Hiroshima, Japan H. Furukawa Matsumoto Dental University, Matsumoto, Nagano, Japan S. Kobayashi Chubu Rosai Hospital, Nagoya, Aichi, Japan H. Shoki · R. Motoya Graduate School of Psychological Science, Health Sciences University of Hokkaido, Sapporo, Hokkaido, Japan H. Shoki National Cancer Center Hospital, Tokyo, Japan R. Motoya Center for Medical Education and Career Development, Fukushima Medical University, Fukushima, Japan M. Saito Division of Pediatric Dentistry, Department of Dental Science, Institute of Personalized Medical Science, Health Sciences University of Hokkaido, Sapporo, Hokkaido, Japan Y. Abiko Division of Oral Medicine and Pathology, Department of Dental Science, Institute of Personalized Medical Science, Health Sciences University of Hokkaido, Sapporo, Hokkaido, Japan Y. Sakano (*) School of Psychological Science, Health Sciences University of Hokkaido, 2-5 Ainosato, Kita-ku, Sapporo, Hokkaido 002-8072, Japan Tel. +81-11-778-7558; Fax +81-11-770-5035 e-mail: [email protected]

izing Scale (PCS), General Self-Efficacy Scale (GSES), and Anxiety Sensitivity Index (ASI) scores in the BMS patients were compared with the scores of Japanese healthy participants (PCS, n = 449; GSES, n = 278; ASI, n = 9603) reported in previous studies. Catastrophizing and anxiety sensitivity were significantly higher in the BMS patients than in the healthy subjects (P < 0.001). In BMS patients, catastrophizing was significantly correlated with pain severity, stressresponse, psychological disability, social disability, and handicap. Cancer phobia was significantly correlated with psychological disability and handicap. Since catastrophizing showed a higher correlation with BMS symptoms than cancer phobia, catastrophizing might be a more significant cognitive factor affecting symptoms than cancer phobia in BMS patients in the Japanese population. Key words Burning mouth syndrome · Pain-related catastrophizing · Cancer phobia · Anxiety sensitivity · Self-efficacy

Introduction Burning mouth syndrome (BMS) is a burning discomfort or pain affecting the soft oral tissues in people with clinically normal oral mucosa.1 This condition mainly affects middleaged or elderly women; the female to male ratio is between 3:1 and 16:1.2,3 The etiology is still unclear, although the proposed etiological factors have been classified as local (oral candidiasis, parafunctional habits, allergy), systemic (diabetes, deficiency of iron, vitamin B12 and folate), psychogenic, and idiopathic.2 The prevalence of patients reporting burning sensations is estimated to be 3.3%–18.3% of the population, whether the etiology is clear or not.4,5 Patients reporting burning sensations with unknown etiology, which generally correspond to a diagnosis of BMS, are reported to make up 0.7%–7.9% of the population.6,7 In Japan, BMS has been reported in approximately 0.8% of patients who visit dental clinics.8 It is advisable for BMS patients to have medical treatment, since in 70% of the patients the symptoms never spontaneously decrease.9,10

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Many treatments for BMS have been attempted, including medication and psychological approaches.11,12 Since psychogenic factors are strongly associated with BMS symptomatology, a psychological treatment approach has often been used, with or without other procedures.13 Among these treatments, cognitive behavior therapy focusing on reducing the dysfunctional cognitive factor is the only psychological approach that has been statistically confirmed as an effective BMS treatment.11,12 Although some studies have confirmed the effectiveness of cognitive behavior therapy for BMS,14,15 it is still unclear what cognitive factor should be the focus of the therapy. Therefore, practitioners require a high level of expertise to identify the dysfunctional cognitive characteristics in BMS patients. If the treatment components of cognitive behavior therapy are designed to focus on the dysfunctional cognitive factors, the treatment period is likely to be shorter. Cancer phobia is a common cognitive characteristic in BMS patients as 45%–74% of BMS patients display cancer phobia,16,17 but whether cancer phobia affects the symptoms of BMS is still unknown. BMS is a chronic pain condition in the oral region. The cognitive factors for patients with chronic pain in other regions have been investigated.18 Self-efficacy, pain-related catastrophizing, and anxiety sensitivity are crucial cognitive factors affecting symptoms of chronic pain.18 Self-efficacy is the belief that one is capable of performing in a certain manner to attain certain goals, which is an important factor in self-management of pain.19 Pain-related catastrophizing is an exaggerated negative orientation toward pain, possibly leading to exacerbation of the pain.20 Anxiety sensitivity refers to a patient’s predisposition to fear anxiety symptoms. Patients with higher anxiety sensitivity may experience a more negative emotional response to pain.21 These cognitive factors may also be important factors in BMS patients. The present study investigated how cognitive characteristics, including cancer phobia, self-efficacy, pain-related catastrophizing, and anxiety sensitivity, affect the symptoms of BMS in the Japanese population.

Methods Participants A total of 46 BMS patients (44 women and 2 men; mean age, 59.89 ± 9.57 years; range, 30–79 years; Table 1) were evaluated at the Department of Medical Psychology of the Health Sciences University of Hokkaido Hospital. The patients were diagnosed with BMS according to the following inclusion criteria: (1) the major complaint of the patient was a burning sensation or pain on the tongue alone, or on both the tongue and oral mucosa; and (2) no local or systemic causes for oral burning22 were founded by detailed clinical evaluation and laboratory tests. Data collection was conducted in 2006–2008. Permission for the study was obtained from the Health Science University of Hokkaido Hospital Ethical Committee. Written consent was obtained from all patients.

Measures Severity of tongue pain. Severity of tongue pain was measured using the Japanese version of the Brief Pain Inventory (BPI) pain severity subscale.23 The BPI pain severity subscale consists of four items (the most intense, weakest, and average pain experienced during the past week, and current pain) using an 11-point Likert scale, where 0 represents “no pain” and 10 represents “pain as bad as you can imagine.” Oral-related quality of life. Oral-related quality of life (QOL) was measured using the Japanese version of the Oral Health Impact Profile-49.24 We used the three subscales of the OHIP, consisting of six “psychological disability” items, six “social disability” items, and five “handicap” items. Respondents were asked to rate the frequency with which they experienced different difficulties in daily life owing to oral problems on a five-point Likert scale, where 1 represents “not at all” and 5 represents “always.”

Table 1. Average scores for cognitive factors in BMS patients and healthy participants

Cancer phobia PCS

GSES ASI

BMS (this study) Average (SD)

Healthy participants (previous studies) Average (SD)

t value

25.56 (28.65) (n = 45; 43 females, 2 males; average age, 59.91 years) 28.19 (9.70) (n = 46; 44 females, 2 males; average age, 59.89 years)





7.30 (3.89) (n = 43; 41 females, 2 males; average age, 59.61 years) 24.81 (9.09) (n = 44; 41 females, 2 males; average age, 59.19 years)

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Matsuoka & Sakano (2006) 22.04 (9.84) (n = 449; 293 females, 153 males; average age, 19.98 years) Sakano & Tohjoh (1986)27 6.58 (3.37) (n = 278; 194 females, 84 males; age range, 18–21 years) Muranaka et al. (2001)28 17.10 (10.53) (n = 9603; 4535 females, 5068 males; average age, 20.4 years)

4.87***

1.15 n.s. 4.40***

BMS, burning mouth syndrome; PCS, Pain Catastrophizing Scale; GSES, General Self-Efficacy Scale; ASI, Anxiety Sensitivity Index; n.s., not significant *** P < 0.001

162 Table 2. Correlations between BMS symptoms and cognitive factors

Pain severity OHIP psychological OHIP social OHIP handicap SRS-18

Cancer phobia

PCS

GSES

ASI

0.09 n.s. 0.30 * 0.24 n.s. 0.31 * 0.20 n.s.

0.56 *** 0.47 ** 0.41 ** 0.63 *** 0.54 ***

0.03 n.s. −0.21 n.s. −0.23 n.s. −0.27 † −0.24 n.s.

0.12 n.s. 0.07 n.s. 0.00 n.s. 0.26 n.s. 0.01 n.s.

OHIP, Oral Health Impact Profile; SRS-18, Stress Response Scale-18 † P < 0.10, * P < 0.05, ** P < 0.01, *** P < 0.001

Stress response. The stress response was measured using the Stress Response Scale-18 (SRS-18).25 The SRS-18 consists of 18 items, and rates the extent of the stress response on a four-point Likert scale, where 0 represents “no stress at all” and 3 represents “high stress.” The SRS-18 has three subscales entitled “depression-anxiety,” “irritability-anger,” and “hopelessness.” Cancer phobia. Patients were asked to answer the question “How much do you fear tongue cancer?” by rating their fear on an 11-point Likert scale, where 0 represents “no fear” and 10 represents “worst fear.” Pain-related catastrophizing. Pain-related catastrophizing was measured using the Japanese version of the Pain Catastrophizing Scale (PCS).26 The PCS consists of 13 items, and respondents were asked to rate the frequency with which they experienced different pain-related thoughts or feelings on a five-point Likert scale, where 0 represents “not at all” and 4 represents “all the time.” Self-efficacy. Self-efficacy was measured using the General Self-Efficacy Scale (GSES).27 The GSES consists of 16 items, and respondents were asked to rate their belief in their competence to cope with a broad range of stressful or challenging demands on a two-point scale (yes or no). Anxiety sensitivity. Anxiety sensitivity was measured using the Japanese version of the Anxiety Sensitivity Index (ASI).28 The ASI consists of 16 items, and respondents were asked to rate the extent to which they found anxiety-related sensations to be catastrophic in outcome on a five-point Likert scale, where 0 represents “very little” and 4 represents “very much.”

Statistical analysis The PCS, GSES, and ASI scores in the BMS patients were compared with the scores of Japanese healthy participants reported in previous studies.26–28 P values were calculated using Student’s t test. Differences between the BMS group and the healthy participants in the previous studies were considered statistically significant when P < 0.05. Correlation analyses were also performed to explore the relationship between symptoms of BMS and cognitive factors.

Results BMS patients participating in this study had experienced burning sensations for an average of 26.28 months (range, 1–78 months). The average pain intensity score was 4.21 on the 11-point Likert scale, indicating that the patients had experienced moderately intense pain. Table 1 shows the mean scores for PCS, GSES, ASI, and cancer phobia in BMS patients and healthy participants. The PCS score differed significantly between the BMS patients and the healthy subjects, indicating that BMS patients exhibited more pain-related catastrophizing. Similarly, BMS patients had higher ASI scores than the healthy subjects. There were no differences in GSES scores between BMS patients and the healthy subjects. Correlation analyses were performed to explore the relationships between pain severity, oral-related QOL, stress responses, and cognitive factors, including cancer phobia, self-efficacy, pain-related catastrophizing, and anxiety sensitivity (Table 2). The cancer phobia score showed no correlation with the pain severity or stress response scores, but it was positively correlated with the scores for psychological disability and handicap (psychological disability, r = 0.30, P < 0.5; handicap, r = 0.31, P < 0.5). The PCS score was significantly correlated with the scores for pain severity, oral-related QOL, and stress response (r = 0.41–0.63, P < 0.01). The ASI and GSES scores showed no significant correlation with any BMS symptoms.

Discussion The present study investigated the effect of cognitive factors, including cancer phobia, pain-related catastrophizing, selfefficacy, and anxiety sensitivity, on BMS symptoms in the Japanese population. Correlations between BMS symptoms and cognitive factors were evaluated. BMS symptoms were significantly correlated with cancer phobia and pain-related catastrophizing. These two cognitive factors may be dysfunctional cognitive parameters in Japanese BMS patients. Interestingly, pain-related catastrophizing showed a higher correlation with BMS symptoms than cancer phobia. Furthermore, pain-related catastrophizing was significantly correlated with oral-related QOL and stress responses. BMS patients with a higher level of pain-related catastrophizing exhibited a lower level of QOL and felt more stressed. Pain-

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related catastrophizing may thus be a more significant cognitive factor affecting symptoms than cancer phobia in BMS patients in the Japanese population. In other types of chronic pain such as neuropathic pain, correlations have been observed between pain-related catastrophizing and disability.29 Our results are consistent with these results. Pain-related catastrophizing predicts future disability in daily life, which might be involved in ongoing pain in chronic pain patients.30 The ongoing pain and pain disability in BMS patients may be influenced by pain-related catastrophizing. Improvement of pain-related catastrophizing might relieve pain and improve the functioning of chronic pain patients.31,32 An intervention resulting in the alteration of pain-related catastrophizing might provide symptomatic relief for BMS patients. In our study, the higher scores for cancer phobia reflected the higher levels of psychological disability and handicap of oral-related QOL. According to Locker’s model of oral health for conceptual domains in the hierarchy of social impact, discomfort or functional limitation caused by diseases leads to physical, psychological, or social disability,33 which then cause handicaps. Cancer phobia was not correlated with pain severity, but it was significantly correlated with the psychological disability and handicap parameters in oral QOL. This result suggests that cancer phobia independently affects the QOL of BMS patients regardless of the severity of the pain. However, the possibility that cancer phobia affects the pain level in some BMS patients cannot be ruled out. A previous study reported that cancer phobia affected the severity of pain in one BMS patient.34 Further investigation is needed to clarify what type of BMS patient has pain levels directly affected by cancer phobia. No correlation between BMS symptoms and anxiety sensitivity was found in the present study. The level of anxiety sensitivity in BMS patients was significantly higher than in the controls. Anxiety sensitivity is defined as the fear of anxiety-related sensations.35 Patients with higher anxiety sensitivity are more likely to manifest these feelings through physical symptoms.36 Symptoms in BMS patients were not related to anxiety sensitivity, although BMS patients may experience somesthesia more acutely than non-BMS subjects. The ASI used in this study included items assessing anxieties not involving somatic symptoms, such as “When I am nervous, I worry that I am mentally ill.” These items might have contributed to the lack of correlation between BMS symptoms and anxiety sensitivity. Another reason for the lack of correlation was that the ASI has no item relating to physical symptoms in the oral region. Further investigation is needed to explore the effect on BMS symptoms of fear of anxiety-related symptoms in the oral region. There was no significant correlation between selfefficacy and BMS symptoms. We used a General SelfEfficacy Scale, which enabled us to establish that general self-efficacy was not associated with BMS symptoms. There is no impairment of function in daily life in BMS patients, although they suffer from burning sensations in the oral region.37 Therefore, the GSES score, which reflects responses toward a broad range of stressful or challenging demands, did not differ between BMS patients and healthy partici-

pants and was not related to the severity of burning sensations in BMS patients. Treatment for other chronic pain conditions, such as musculoskeletal pain, often starts with techniques aimed at improving the patient’s functioning in daily life. These techniques may be ineffective in BMS. There are some limitations to our study. First, although we compared the scores of cognitive factors in BMS patients to scores of healthy subjects, age and the male : female ratio were not matched between the BMS patients and the healthy subjects. Further research is needed to compare cognitive profiles in BMS patients with age- and sexmatched healthy subjects. Second, since we adopted a crosssectional design in this study, it was difficult to determine whether cognitive factors causally influenced BMS symptoms. Therefore, we need to conduct a study with a longitudinal design. The present study demonstrated for the first time that pain-related catastrophizing directly affects BMS symptoms in the Japanese population. This result indicates that BMS patients might be treated by using pain catastrophizing as the treatment target. If treatment focusing on pain-related catastrophizing is successful, a program could be developed clearly outlining the treatment components, which might lead to a shortening of the treatment period. Further investigation is needed to explore the effects of treating BMS patients with a regime focusing on pain-related catastrophizing.

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