Medically Unexplained Oropharyngeal Dysphagia at the University Hospital ENT Outpatient Clinic for Dysphagia: A Cross-Sectional Cohort Study

Medically Unexplained Oropharyngeal Dysphagia at the University Hospital ENT Outpatient Clinic... Medically unexplained oropharyngeal dysphagia (MUNOD) is a rare condition. It presents without demonstrable abnor- malities in the anatomy of the upper aero-digestive tract and/or swallowing physiology. This study investigates whether MUNOD is related to affective or other psychiatric conditions. The study included patients with dysphagic complaints who had no detectible structural or physiological abnormalities upon swallowing examination. Patients with any underlying disease or disorder that could explain the oropharyngeal dysphagia were excluded. All patients underwent a standardized examination protocol, with FEES examination, the Hospital Anxiety and Depression Scale (HADS), and the Dysphagia Severity Scale (DSS). Two blinded judges scored five different FEES variables. None of the 14 patients included in this study showed any structural or physiological abnormalities during FEES examination. However, the majority did show abnormal piecemeal deglutition, which could be a symptom of MUNOD. Six patients (42.8%) had clinically relevant symptoms of anxiety and/or depression. The DSS scores did not differ significantly between patients with and without affective symptoms. Affective symptoms are common in patients with MUNOD, and their psychiatric conditions could possibly be related to their swallowing problems. Keywords Dysphagia  Affective symptoms  Anxiety  Depression Introduction Patients with swallowing problems are commonly seen at the otorhinolaryngology outpatient clinic. Their oropha- & Rob J.C.G. Verdonschot ryngeal dysphagia (OD) may be attributed to somatic eti- RJCG.Verdonschot@alumni.maastrichtuniversity.nl ologies such as head and neck cancer, progressive Department of Otorhinolaryngology, Head and Neck neurological disorders, or stroke [1–3]. These disorders Surgery, Maastricht University Medical Center, may change the normal anatomy and/or disturb normal PO Box 5800, 6202 AZ Maastricht, The Netherlands function of the upper aero-digestive tract and thereby Emergency Department, Erasmus Medical Center, hamper normal swallowing. Rarely, OD occurs without Rotterdam, The Netherlands demonstrable abnormalities in the anatomy of the upper School of Mental Health and Neurosciences (MHeNS), aero-digestive tract and/or swallowing physiology, Maastricht University, Maastricht, The Netherlands prompting a diagnosis of medically unexplained oropha- GROW-School for Oncology and Developmental Biology, ryngeal dysphagia (MUNOD) [4]. In the literature, this Maastricht University Medical Center, Maastricht, condition is known by various names: functional dyspha- The Netherlands gia, swallowing phobia, psychogenic dysphagia, or Department of Methodology and Statistics, CAPHRI, phagophobia [4]. A functional somatic disorder is defined Maastricht University, Maastricht, The Netherlands as physical complaints or symptoms impairing normal Department of Psychiatry and Psychology, Maastricht function of the bodily process that are not attributable to an University Medical Center, Maastricht, The Netherlands 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia underlying structural disease [5]. Functional somatic dis- integrated (otorhinolaryngological and psychiatric) man- orders and comorbid anxiety and depression are both agement strategies in the context of best clinical practice. associated with increased severity of symptoms and greater illness burden [6]. Medical specialties tend to apply their own diagnostic labels to functional somatic disorders. Materials and Methods Psychiatry uses the term somatic symptom disorder, while other specialties make their own specific diagnosis (e.g., Patients irritable bowel syndrome (IBS), fibromyalgia (FM), func- tional dyspepsia (FD)) [5, 7]. In the field of mental health, Patients with OD complaints (usually choking) who were patients with MUNOD are frequently diagnosed with a referred to the outpatient clinic for dysphagia of the functional somatic disorder or rarely with phagophobia Maastricht University Medical Center (MUMC?) between (fear of swallowing). According to the DSM-V classifica- July 2011 and April 2016, without detectible abnormalities tion, phagophobia belongs to the category of ‘specific in swallowing examination, were included in the study. phobias’ [7], whereby exposure to the phobic stimulus The following exclusion criteria were applied: age younger provokes an immediate anxiety response. The phobic sit- than 18, age older than 85 (presbyphagia), complaints of uation is avoided or endured with intense distress. Also, the esophageal dysphagia (e.g., swallowing-related chest pain, specific phobia interferes with a patient’s normal routine, esophageal regurgitation, history of esophageal dysphagia), functioning, or social activities. Phagophobia can only be history of head and neck cancer, evidence or suspicion of diagnosed if other psychiatric or somatic conditions are neurodegenerative disease (e.g., Myasthenia Gravis, mul- excluded as a possible cause for the dysphagia and tiple sclerosis, Parkinson’s disease), stroke patients, accompanying emotional and bodily distress [7]. Patients patients with a Zenker’s diverticulum or cervical spine with phagophobia experience an abnormal sensation during abnormalities, patients with any other somatic disease or swallowing, sometimes accompanied by behavioral disorder that could explain the OD complaints, a score abnormalities during swallowing examination [7]. In the below 23 on the Mini Mental State Examination (MMSE) literature, phagophobia is often described in children [8, 9], [15], or not knowing the Dutch language. Informed consent but little is known about this condition in adults. Given the was obtained from all patients. strong association of medically unexplained symptoms with affective conditions, it is advisable to use the broader Examination Protocol term ‘MUNOD’ (instead of ‘phagophobia’). It may be a symptom within other psychiatric conditions like obses- All patients underwent a standardized examination proto- sive-compulsive disorder, panic disorder, post-traumatic col (prospectively collected data) used in daily clinical stress disorder (PTSD), social phobia, or depression [10]. practice at the outpatient clinic for dysphagia. This proto- In patients with persistent complaints of MUNOD who do col comprises a structured interview, standardized otorhi- not show detectible abnormalities upon swallowing nolaryngology examination, a standardized FEES examination performed with fiberoptic endoscopic evalu- examination [16], the Hospital Anxiety and Depression ation of swallowing (FEES) or videofluoroscopic swal- Scale (HADS) [17], a dysphagia severity scale (DSS) lowing study (VFSS), and who do not present with an [14, 18], Body Mass Index (BMI) measurement, and the underlying somatic disease, a possible cause of the com- MMSE [15]. The FEES-examinations were carried out by plaints should be sought in a psychiatric condition (e.g., an experienced laryngologist together with the speech somatic symptom disorder, phagophobia, affective disor- therapist. First, patients had to perform three swallows of der, PTSD) [3, 4, 11, 12]. In most complex and high-uti- 10 cc thin liquid (water), then three swallows of 10 cc lizing patients with OD, affective or somatoform standardized applesauce (One 2 fruit ) (hereafter ‘thick comorbidity should therefore be considered [13, 14]. liquid’), and then one bite-sized cracker (80 gr Delhaize Mini Toast ). All liquids were dyed with 5% methylene Aim blue (10 mg/ml). A flexible fiberoptic endoscope, Pentax FNL-10RP3 (Pentax Canada Inc., Mississauga, Ontario, So far, no other studies have investigated whether patients Canada), was used during the FEES examination. The tip with MUNOD have clinically relevant symptoms of anxi- of the endoscope was in ‘high position,’ just above the ety and depression. This study is the first to inquire whether epiglottis, so the scope could not interfere with closure of MUNOD is related to an affective condition or presents as the laryngeal vestibule [16]. The FEES videos were a symptom within another psychiatric condition. The aim obtained with the Xion SD camera, Xion EndoSTROBE of this study is to better understand the psychiatric symp- camera control unit (PAL 25 fps), and Matrix DS data toms in patients with MUNOD and to provide guidance for station with DIVAS software (Xion Medical, Berlin, 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Germany) and recorded on a DVD. Second, the investi- coefficient. Results were expressed as the median (range) gators administered the HADS, a validated tool to assess for continuous variables, while frequencies and proportions clinically relevant symptoms of anxiety and/or depression. (%) were used for ordinal FEES variables. The Mann– It consists of 14 items: seven on the anxiety subscale and Whitney U test and the Chi-squared test were used for seven on the depression subscale. Each single item is group comparisons. Spearman’s rho was used for correla- scored from 0 to 3, resulting in a minimum of 0 and a tions between continuous variables. All statistical analyses maximum of 21 points on each subscale. A higher score were performed with IBM SPSS Statistics for Mac, version indicates more anxiety or depression symptoms. A score of 22.0 (Armonk, NY: IBM Corp.). C 8 on a subscale implies the presence of clinically rele- vant anxiety or depression symptoms, which is an indicator of an anxiety disorder or depression [17, 19, 20]. Third, a Results patient’s subjective swallowing assessment was measured with the DSS, a visual analog scale (VAS); this instrument Participants is a psychometric response scale for measuring subjective characteristics or attitudes [14, 18]. Dysphagic patients Approximately 120 patients per year visited the outpatient specify their level of agreement with a statement or ques- clinic for dysphagia. Patients were referred by general tion by indicating a position along a continuous line practitioners, otorhinolaryngologists, or other specialists between two end-points for the DSS. The single question such as a neurologist or pulmonologist. The main reason was, ‘‘How do you rate your swallowing today?’’ A score for referral was to exclude pathology of the upper aero- of 100 (maximum) indicates normal swallowing. The digestive tract as a cause for OD. Fourteen patients met the MMSE is a tool to screen patient’s cognitive status. A score criteria for MUNOD and were included in the study. The below 23 is interpreted as mild cognitive impairment for median age was 52 (19–68). In total seven of the partici- which a formal cognitive assessment to determine the pants (50%) were female. See Table 1 for general patients’ pattern and extent of deficits is recommended. Therefore, characteristics. to reduce possible bias in the HADS and DSS outcomes due to cognitive dysfunctions in the present study, patients Observer Agreement with an MMSE below 23 were excluded. Table 2 shows levels of inter- and intraobserver agreement FEES Variables for all FEES variables with 95% confidence interval. Intraobserver agreement levels are shown for both raters To be sure that none of the selected patients had severe separately. All levels of agreement were almost perfect abnormalities during FEES examination (e.g., severe (Kappa [ 0.9). The lowest level of interobserver agree- pooling, deep penetration, aspiration), suggesting a possi- ment was 0.95 (95% CI 0.89–1.00) for postswallow pyri- ble underlying somatic cause, five visuoperceptual ordinal form sinus pooling. The lowest level of intraobserver variables (piecemeal deglutition, postswallow vallecular agreement was 0.90 (95% CI 0.80–1.00) for postswallow pooling, postswallow pyriform sinus pooling, laryngeal vallecular pooling. The prevalence of impairment was very penetration, and aspiration) were scored by two indepen- low for all variables. dent judges [13, 21–26]. All of these variables were scored for every FEES swallow at varying speed. The judges FEES Variables underwent consensus training for these measurements, as described previously [13, 21–26]. Both judges were blin- Descriptive data of the FEES variables are displayed in ded to the patients’ identity and medical history. The jud- Table 3. Piecemeal deglutition was rated as normal (cate- ges were also blinded to each other’s scores. To determine gory 0) in 31.0% (N = 13), 16.7% (N = 7), and 7.1% intraobserver agreement, 30 (29%) of the FEES swallows (N = 1) of the swallows for thin liquid consistency, thick were rated twice (repeated measurements). These FEES liquid consistency, and bite-sized cracker, respectively. In swallows were randomly selected and again blinded for five patients, postswallow vallecular pooling was rated as both judges. Fatigue-related observer bias was avoided by mild (14.3 and 7.1% of the swallows for thin liquid and limiting the judge’s rating task to two hours per session. thick liquid consistency, respectively), but in none of these patients was pooling observed in all seven recorded swal- Statistical Analysis lows. All five patients showed at least one normal swallow without vallecular pooling. None of the swallows was rated Levels of interobserver and intraobserver agreement were as severe vallecular pooling (category 2). Penetration was measured for each variable by the linear weighted kappa observed in two patients. The first patient showed a trace of 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Table 1 Patients’ characteristics Subject Age Gender BMI MMSE- Psychiatric history Psychiatric Referred by No. of visits score medication otorhinolaryngology outpatient clinic MUMC? 1 56 Female 29 30 – – GP 6 2 27 Male 17 30 Pervasive developmental – Internist 1 disorder—not otherwise specified 3 43 Male 17 25 Cluster B personality Temazepam, GP 2 disorder Oxazepam 4 41 Female 21 23 Panic disorder Citalopram Otorhinolaryngologist 3 5 51 Male 23 30 – – MV 1 6 68 Male 25 29 – – GP 1 7 26 Male MV 23 – – GP 9 8 53 Male MV 23 – – Otorhinolaryngologist 3 9 63 Female MV 26 – – GP 1 10 19 Female 16 23 – – GP 1 11 60 Female 37 29 Psychotic depression Quetiapine Neurologist 1 12 61 Female 34 29 – – Internist 1 13 34 Female 20 30 – – GP 2 14 66 Male 25 30 – – Pulmonologist 2 BMI Body Mass Index, MMSE mini mental state examination, GP general practitioner, MV missing value, MUMC? Maastricht University Medical Center methylene blue on the laryngeal side of the epiglottis DSS during the first thin liquid swallow. The second patient showed deeper penetration, near the vocal folds, in multi- The median score for the DSS was 66.0 (18–100). Spear- ple swallows and was therefore excluded because an man’s rho revealed no significant correlation between age underlying somatic cause of OD could not be excluded. and DSS. The DSS was not significantly different for None of the patients showed aspiration or pyriform sinus patients with clinically relevant symptoms of anxiety or pooling during the swallowing examination. The study depression compared to patients without symptoms of population was too small to perform further statistical anxiety or depression. Males scored significant higher on analyses. the DSS compared to females. See Table 4 for the results of the Mann–Whitney U tests for group comparison. Hads Discussion Six of the 14 participants (42.8%) showed clinically rel- evant symptoms of anxiety (score C 8 on the anxiety This is the first study that investigates swallowing function subscale). Three of the 14 (21.4%) showed clinically in relation to symptoms of anxiety and depression in relevant symptoms of depression (score C 8 on the patients with MUNOD. All 14 included patients presented depression subscale). These three also had a score C 8on with complaints of OD, and none showed structural the anxiety subscale. Thus, 42.8% (N = 6) of the partic- abnormalities during FEES examination. However, the ipants had clinically relevant symptoms of anxiety and/or majority showed abnormal piecemeal deglutition, which depression. The Chi-squared test showed no gender dif- could be an early symptom of an underlying somatic dis- ferences between patients with and without clinically order impairing normal swallowing physiology. However, relevant symptoms of anxiety (p = 0.28) or depression it is conceivable that abnormal piecemeal deglutition is a (p = 0.51). The Mann–Whitney U test showed no age clinically relevant symptom of MUNOD. Since these differences between patients with and without clinically patients are often anxious about swallowing, multiple relevant symptoms of anxiety (p = 1.00) or depression swallows of smaller fragments of the same bolus may offer (p = 0.76). them a sense of safety or control. In these patients, 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Table 2 Interobserver and intraobserver agreement levels per FEES variable assessed with linear weighted Kappa and 95% confidence interval FEES outcome Definition Ordinal scale Interobserver Intraobserver agreement (95% CI) variable agreement (95% Observer 1 Observer 2 CI) Piecemeal Sequential swallowing on the Five-point scale (0–4) 0.99 (0.97–1.00) 0.93 (0.84–1.00) 0.93 (0.84–1.00) deglutition same bolus 0 = no additional swallows 1 = one additional swallow 2 = two additional swallows 3 = three additional swallows 4 = four additional swallows Postswallow Pooling in valleculae after the Three-point scale (0–2) 0.95 (0.91–1.00) 0.96 (0.89–1.00) 0.90 (0.80–1.00) vallecular swallow 0 = no pooling pooling 1 = filling of less than 50% of the valleculae 2 = filling of more than 50% of the valleculae Postswallow Pooling in pyriform sinuses after Three-point scale (0–2) 0.95 (0.89–1.00) 1.00 1.00 pyriform sinus the swallow 0 = no pooling pooling 1 = trace to moderate pooling 2 = severe pooling up to complete filling of the sinuses Penetration and Penetration of bolus in the Three-point scale (0–2) 0.98 (0.96–1.00) 0.97 (0.90–1.00) 0.97 (0.90–1.00) laryngeal vestibule, above the aspiration 0 = no penetration vocal folds 1 = penetration Aspiration of bolus below the 2= aspiration vocal folds Results of intraobserver agreement are given for both observers Lower scores refer to normal functioning, whereas higher scores refer to more severe disability Kappa values: \ 0 = less than chance agreement; 1 = perfect agreement piecemeal deglutition seems to be a habitual coping strat- [28]. The bladder–gut–brain axis is an interesting frame- egy rather than a subclinical neurogenic impaired swal- work. It suggests a bidirectional pathway between brain lowing pattern. Nevertheless, follow-up for a possible and body, assuming that both functional and affective progressive neurologic disease is recommended. It is disorders are stress related and that functional symptoms assumed that swallowing physiology in patients with are a sensitized response to earlier threats. This sensitiza- MUNOD is normal. However, an interesting question is tion might mediate false-alarm signals (alarm falsification whether MUNOD could disturb normal swallowing phys- as a defense system). That, in turn, could provoke emo- iology. Roland et al. evaluated the incidence of esophageal tional and physical distress, resulting in psychiatric con- contractility disturbances in psychiatric patients [27]. ditions and functional disorders like MUNOD [6, 28]. A Manometry showed a high percentage of functional motor study by Dum et al. raised the possibility that motor areas impairment in patients with complaints of anxiety and/or of the cerebral cortex are important in the stress and depression, while endoscopy in these patients showed no depression connectome [29], and Grillon et al. suggested structural abnormalities [27]. In a large prospective popu- that anxiety increases motor response inhibition [30]. lation-based study, Koloski et al. showed that anxiety is an These studies indicate a relationship between affective independent predictor for new onset functional gastroin- function and motor function and thus strengthen the testinal disorders like irritable bowel syndrome, suggesting assumption that functional complaints might be part of a that affective disorders can underlie physical symptoms hypersensitivity or alarm-falsification disorder [6]. By 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Table 3 Frequency distribution FEES category frequencies of swallows per category of the different FEES variables, given Thin liquid consistency Thick liquid consistency Bite-sized cracker as absolute numbers (N) and N (%) N (%) N (%) percentages (%) N =42 N =42 N =14 Piecemeal deglutition Category 0 13 (31.0) 7 (16.7) 1 (7.1) Category 1 10 (23.8) 15 (35.7) 2 (14.3) Category 2 13 (30.9) 10 (23.8) 2 (14.3) Category 3 1 (2.4) 3 (7.1) 2 (14.3) Category 4 5 (11.9) 6 (14.3) 6 (42.9) MV 0 1 (2.4) 1 (7.1) Postswallow vallecular pooling Category 0 35 (83.3) 36 (85.7) 12 (85.7) Category 1 6 (14.3) 3 (7.1) 0 Category 2 0 0 0 MV 1 (2.4) 3 (7.1) 2 (14.3) Postswallow pyriform sinus pooling Category 0 41 (97.6) 40 (95.2) 12 (85.7) Category 1 0 0 0 Category 2 0 0 0 MV 1(2.4) 2 (4.8) 2 (14.3) Penetration/aspiration Category 0 40 (95.2) 41 (97.6) 12 (85.7) Category 1 1 (2.4) 0 0 Category 2 0 0 0 MV 1 (2.4) 1 (2.4) 2 (14.3) Missing value; FEES variable could not be rated deglutition). So far, no studies have been published on this Table 4 Comparison of DSS between patients with clinically relevant symptoms of anxiety or depression and patients without symptoms of subject. However, the assumption that patients with anxiety or depression using Mann–Whitney U test and comparison of MUNOD must have a normal swallowing function might DSS between male and female patients using the Mann–Whitney be incorrect. Through this bidirectional pathway, a psy- U test chiatric problem can have sensorimotor effects on the N DSS score Level of significance swallowing function without there being any other cause of Median (range) p-value dysphagia, such as a chronic neurological disorder. Then, it HADS-D C 8 3 85.0 (18–100) 0.659 would be plausible that OD can be caused by affective HADS-D \ 8 11 57.0 (31–98) disorders or psychiatric conditions, even when the swal- HADS-A C 8 6 76.0 (18–100) 0.662 lowing physiology is disturbed. In this study, none of the HADS-A \ 8 8 55.5 (31–98) participants had symptoms indicating an underlying Male 7 85.0 (44–100) 0.017 somatic disease, and none showed other abnormalities Female 7 54.0 (18–77) during structured interviews or general otorhinolaryngol- ogy examination (normal cranial nerve integrity, speech, DSS Dysphagia Severity Score, HADS-D Depression subscale of the etc.). Although a somatic cause of dysphagia might seem Hospital Anxiety and Depression Scale, HADS-A Anxiety subscale of the Hospital Anxiety and Depression Scale unlikely, MUNOD should always be a diagnosis of Statistically significant exclusion. Previous research showed a high prevalence of clinically relevant affective symptoms in OD patients [13, 14, 31]. The present study underpins these data. It also shows a high implication, MUNOD and functional motor impairment prevalence (42.8%) of clinically relevant affective symp- may be interrelated too, causing disturbances of the normal toms, which indicates that MUNOD seems to be related to swallowing physiology (such as increased piecemeal 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia affective conditions in more than 40% of the cases. Four of prolonged dysphagic complaints, with no indication of a the participants (28.5%) had already been diagnosed with a somatic disease or abnormality, psychiatric conditions psychiatric condition (psychotic depression, panic disorder, must be considered as a possible cause of OD. Validated pervasive developmental disorder—not otherwise speci- psychological screening questionnaires could be helpful in fied, cluster B personality disorder). The patient with the detection of affective conditions but also of other cluster B personality disorder showed clinically relevant psychiatric conditions. Involvement of a psychiatrist and/or symptoms of anxiety and depression, and the patient with psychologist is recommended. panic disorder exhibited clinically relevant symptoms of anxiety. In these patients, MUNOD and affective symp- Limitations of the Study toms are likely to be part of their psychiatric disorder. The DSS scores were not significantly different between This investigation has some limitations. First, since patients with and without clinically relevant affective MUNOD is a rare condition, the number of patients symptoms. Apparently, clinically relevant symptoms of included in the study is small, so only a limited statistical anxiety and depression are not related to the severity of analysis could be performed. Second, the HADS ques- MUNOD symptoms. A psychological screening question- tionnaire was used for screening of anxiety and depression naire, like the HADS, is a simple tool for the preliminary symptoms. Possibly, a different screening tool or multiple assessment of the affective state of a patient. However, the screening tools would have led to different results. Third, expertise of a psychiatrist is essential to a definitive diag- three of the participants were taking psychiatric medication nosis and treatment of any psychiatric condition, including (see Table 1), which could have a negative effect on phagophobia or other anxiety disorders, and depression. It swallowing [32, 33]. Furthermore, the use of psychiatric might be helpful to draw upon the patient’s psychiatric medication could have led to an underestimation of the history and to involve his or her own psychiatrist when HADS scores. Furthermore, this investigation used a cross- preparing a multidisciplinary treatment strategy. Involve- sectional study design and was not intended as a therapy- ment of a psychiatrist would obviously be necessary. effect study; the effect of different treatment options could However, the patient must be willing to cooperate and be examined in future research, which could also specify accept that a psychiatric problem might be the cause of the treatment strategies in patients with MUNOD and psychi- swallowing problems. In this study, only four patients atric comorbidity. could be convinced to visit a psychiatrist after visiting the outpatient clinic for dysphagia. Following referral to the psychiatrist, one patient was diagnosed with an anxiety Conclusion disorder and one patient was diagnosed with an identity disorder. Two of the referred patients were already known MUNOD is a rare condition that is difficult to diagnose. with a psychiatric disorder (panic disorder and psychotic We hope to help dysphagia caregivers by sharing our depression), see Table 1. Early recognition of MUNOD results and experiences. Patients deserve a professional and a motivational trajectory towards integrated care are approach, particularly because their diagnostic trajectory necessary to develop effective treatment strategies, to has often been long and inconclusive. Affective symptoms reduce health care consumption and health care costs, to are common in these patients. MUNOD could be a symp- decrease the risk of iatrogenic damage arising from con- tom of a psychiatric condition or part of the alarm falsifi- tinuous diagnostic intervention, and to prevent frustration cation defense system, suggesting that physical symptoms in the interaction between physician and patient [11]. and affective disorders are stress-related and a response to Almost all of the participants had already consulted mul- earlier threats. Consultation of a psychiatrist for patients tiple specialists or had made recurrent visits to outpatient with MUNOD is recommended as part of a pathway toward clinics all over the Netherlands. Consultation of a psychi- multidisciplinary integrated care. atrist must be considered as an early option in the diag- nostic strategy of MUNOD instead of the ‘last resort’ after unsuccessful treatment. Diagnosis and treatment of an Compliance with Ethical Standards underlying psychiatric disease may improve the swallow- ing problems. It is important to realize that affective Conflicts of interest The authors declare that they have no conflict of symptoms are frequently present in patients with MUNOD. interest (financial or non-financial). Assuming a bidirectional pathway between brain and body, Ethical Approval The authors declare that this manuscript has not MUNOD could be understood as a symptom of physical been submitted to other journals and that this manuscript has not been distress or part of an alarm falsification and defense reac- published previously. Consent to submit was received from all co- tion as seen in other functional syndromes. In patients with 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia authors, and all co-authors contributed sufficiently to the scientific oropharyngeal dysphagia. J Psychosom Res. 2013;75(5):451–5. work. Informed consent was obtained from all patients included in https://doi.org/10.1016/j.jpsychores.2013.08.021. this study. 15. Folstein MF, Folstein SE, McHugh PR. ‘‘Mini-mental state’’. A practical method for grading the cognitive state of patients for the clinician. J Psychiatr Res. 1975;12(3):189–98. 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Bernd Kremer MD, PhD 32. Gallagher L, Naidoo P. Prescription drugs and their effects on swallowing. Dysphagia. 2009;24(2):159–66. https://doi.org/10. Carsten Leue MD, PhD 1007/s00455-008-9187-7. 33. Stegemann S, Gosch M, Breitkreutz J. Swallowing dysfunction and dysphagia is an unrecognized challenge for oral drug therapy. Int J Pharm. 2012;430(1–2):197–206. Rob J.C.G. Verdonschot MD Laura W.J. Baijens MD, PhD Sophie Vanbelle MSc, PhD Michelle Florie MD http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Dysphagia Springer Journals

Medically Unexplained Oropharyngeal Dysphagia at the University Hospital ENT Outpatient Clinic for Dysphagia: A Cross-Sectional Cohort Study

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Medicine & Public Health; Otorhinolaryngology; Imaging / Radiology; Gastroenterology; Hepatology
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Abstract

Medically unexplained oropharyngeal dysphagia (MUNOD) is a rare condition. It presents without demonstrable abnor- malities in the anatomy of the upper aero-digestive tract and/or swallowing physiology. This study investigates whether MUNOD is related to affective or other psychiatric conditions. The study included patients with dysphagic complaints who had no detectible structural or physiological abnormalities upon swallowing examination. Patients with any underlying disease or disorder that could explain the oropharyngeal dysphagia were excluded. All patients underwent a standardized examination protocol, with FEES examination, the Hospital Anxiety and Depression Scale (HADS), and the Dysphagia Severity Scale (DSS). Two blinded judges scored five different FEES variables. None of the 14 patients included in this study showed any structural or physiological abnormalities during FEES examination. However, the majority did show abnormal piecemeal deglutition, which could be a symptom of MUNOD. Six patients (42.8%) had clinically relevant symptoms of anxiety and/or depression. The DSS scores did not differ significantly between patients with and without affective symptoms. Affective symptoms are common in patients with MUNOD, and their psychiatric conditions could possibly be related to their swallowing problems. Keywords Dysphagia  Affective symptoms  Anxiety  Depression Introduction Patients with swallowing problems are commonly seen at the otorhinolaryngology outpatient clinic. Their oropha- & Rob J.C.G. Verdonschot ryngeal dysphagia (OD) may be attributed to somatic eti- RJCG.Verdonschot@alumni.maastrichtuniversity.nl ologies such as head and neck cancer, progressive Department of Otorhinolaryngology, Head and Neck neurological disorders, or stroke [1–3]. These disorders Surgery, Maastricht University Medical Center, may change the normal anatomy and/or disturb normal PO Box 5800, 6202 AZ Maastricht, The Netherlands function of the upper aero-digestive tract and thereby Emergency Department, Erasmus Medical Center, hamper normal swallowing. Rarely, OD occurs without Rotterdam, The Netherlands demonstrable abnormalities in the anatomy of the upper School of Mental Health and Neurosciences (MHeNS), aero-digestive tract and/or swallowing physiology, Maastricht University, Maastricht, The Netherlands prompting a diagnosis of medically unexplained oropha- GROW-School for Oncology and Developmental Biology, ryngeal dysphagia (MUNOD) [4]. In the literature, this Maastricht University Medical Center, Maastricht, condition is known by various names: functional dyspha- The Netherlands gia, swallowing phobia, psychogenic dysphagia, or Department of Methodology and Statistics, CAPHRI, phagophobia [4]. A functional somatic disorder is defined Maastricht University, Maastricht, The Netherlands as physical complaints or symptoms impairing normal Department of Psychiatry and Psychology, Maastricht function of the bodily process that are not attributable to an University Medical Center, Maastricht, The Netherlands 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia underlying structural disease [5]. Functional somatic dis- integrated (otorhinolaryngological and psychiatric) man- orders and comorbid anxiety and depression are both agement strategies in the context of best clinical practice. associated with increased severity of symptoms and greater illness burden [6]. Medical specialties tend to apply their own diagnostic labels to functional somatic disorders. Materials and Methods Psychiatry uses the term somatic symptom disorder, while other specialties make their own specific diagnosis (e.g., Patients irritable bowel syndrome (IBS), fibromyalgia (FM), func- tional dyspepsia (FD)) [5, 7]. In the field of mental health, Patients with OD complaints (usually choking) who were patients with MUNOD are frequently diagnosed with a referred to the outpatient clinic for dysphagia of the functional somatic disorder or rarely with phagophobia Maastricht University Medical Center (MUMC?) between (fear of swallowing). According to the DSM-V classifica- July 2011 and April 2016, without detectible abnormalities tion, phagophobia belongs to the category of ‘specific in swallowing examination, were included in the study. phobias’ [7], whereby exposure to the phobic stimulus The following exclusion criteria were applied: age younger provokes an immediate anxiety response. The phobic sit- than 18, age older than 85 (presbyphagia), complaints of uation is avoided or endured with intense distress. Also, the esophageal dysphagia (e.g., swallowing-related chest pain, specific phobia interferes with a patient’s normal routine, esophageal regurgitation, history of esophageal dysphagia), functioning, or social activities. Phagophobia can only be history of head and neck cancer, evidence or suspicion of diagnosed if other psychiatric or somatic conditions are neurodegenerative disease (e.g., Myasthenia Gravis, mul- excluded as a possible cause for the dysphagia and tiple sclerosis, Parkinson’s disease), stroke patients, accompanying emotional and bodily distress [7]. Patients patients with a Zenker’s diverticulum or cervical spine with phagophobia experience an abnormal sensation during abnormalities, patients with any other somatic disease or swallowing, sometimes accompanied by behavioral disorder that could explain the OD complaints, a score abnormalities during swallowing examination [7]. In the below 23 on the Mini Mental State Examination (MMSE) literature, phagophobia is often described in children [8, 9], [15], or not knowing the Dutch language. Informed consent but little is known about this condition in adults. Given the was obtained from all patients. strong association of medically unexplained symptoms with affective conditions, it is advisable to use the broader Examination Protocol term ‘MUNOD’ (instead of ‘phagophobia’). It may be a symptom within other psychiatric conditions like obses- All patients underwent a standardized examination proto- sive-compulsive disorder, panic disorder, post-traumatic col (prospectively collected data) used in daily clinical stress disorder (PTSD), social phobia, or depression [10]. practice at the outpatient clinic for dysphagia. This proto- In patients with persistent complaints of MUNOD who do col comprises a structured interview, standardized otorhi- not show detectible abnormalities upon swallowing nolaryngology examination, a standardized FEES examination performed with fiberoptic endoscopic evalu- examination [16], the Hospital Anxiety and Depression ation of swallowing (FEES) or videofluoroscopic swal- Scale (HADS) [17], a dysphagia severity scale (DSS) lowing study (VFSS), and who do not present with an [14, 18], Body Mass Index (BMI) measurement, and the underlying somatic disease, a possible cause of the com- MMSE [15]. The FEES-examinations were carried out by plaints should be sought in a psychiatric condition (e.g., an experienced laryngologist together with the speech somatic symptom disorder, phagophobia, affective disor- therapist. First, patients had to perform three swallows of der, PTSD) [3, 4, 11, 12]. In most complex and high-uti- 10 cc thin liquid (water), then three swallows of 10 cc lizing patients with OD, affective or somatoform standardized applesauce (One 2 fruit ) (hereafter ‘thick comorbidity should therefore be considered [13, 14]. liquid’), and then one bite-sized cracker (80 gr Delhaize Mini Toast ). All liquids were dyed with 5% methylene Aim blue (10 mg/ml). A flexible fiberoptic endoscope, Pentax FNL-10RP3 (Pentax Canada Inc., Mississauga, Ontario, So far, no other studies have investigated whether patients Canada), was used during the FEES examination. The tip with MUNOD have clinically relevant symptoms of anxi- of the endoscope was in ‘high position,’ just above the ety and depression. This study is the first to inquire whether epiglottis, so the scope could not interfere with closure of MUNOD is related to an affective condition or presents as the laryngeal vestibule [16]. The FEES videos were a symptom within another psychiatric condition. The aim obtained with the Xion SD camera, Xion EndoSTROBE of this study is to better understand the psychiatric symp- camera control unit (PAL 25 fps), and Matrix DS data toms in patients with MUNOD and to provide guidance for station with DIVAS software (Xion Medical, Berlin, 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Germany) and recorded on a DVD. Second, the investi- coefficient. Results were expressed as the median (range) gators administered the HADS, a validated tool to assess for continuous variables, while frequencies and proportions clinically relevant symptoms of anxiety and/or depression. (%) were used for ordinal FEES variables. The Mann– It consists of 14 items: seven on the anxiety subscale and Whitney U test and the Chi-squared test were used for seven on the depression subscale. Each single item is group comparisons. Spearman’s rho was used for correla- scored from 0 to 3, resulting in a minimum of 0 and a tions between continuous variables. All statistical analyses maximum of 21 points on each subscale. A higher score were performed with IBM SPSS Statistics for Mac, version indicates more anxiety or depression symptoms. A score of 22.0 (Armonk, NY: IBM Corp.). C 8 on a subscale implies the presence of clinically rele- vant anxiety or depression symptoms, which is an indicator of an anxiety disorder or depression [17, 19, 20]. Third, a Results patient’s subjective swallowing assessment was measured with the DSS, a visual analog scale (VAS); this instrument Participants is a psychometric response scale for measuring subjective characteristics or attitudes [14, 18]. Dysphagic patients Approximately 120 patients per year visited the outpatient specify their level of agreement with a statement or ques- clinic for dysphagia. Patients were referred by general tion by indicating a position along a continuous line practitioners, otorhinolaryngologists, or other specialists between two end-points for the DSS. The single question such as a neurologist or pulmonologist. The main reason was, ‘‘How do you rate your swallowing today?’’ A score for referral was to exclude pathology of the upper aero- of 100 (maximum) indicates normal swallowing. The digestive tract as a cause for OD. Fourteen patients met the MMSE is a tool to screen patient’s cognitive status. A score criteria for MUNOD and were included in the study. The below 23 is interpreted as mild cognitive impairment for median age was 52 (19–68). In total seven of the partici- which a formal cognitive assessment to determine the pants (50%) were female. See Table 1 for general patients’ pattern and extent of deficits is recommended. Therefore, characteristics. to reduce possible bias in the HADS and DSS outcomes due to cognitive dysfunctions in the present study, patients Observer Agreement with an MMSE below 23 were excluded. Table 2 shows levels of inter- and intraobserver agreement FEES Variables for all FEES variables with 95% confidence interval. Intraobserver agreement levels are shown for both raters To be sure that none of the selected patients had severe separately. All levels of agreement were almost perfect abnormalities during FEES examination (e.g., severe (Kappa [ 0.9). The lowest level of interobserver agree- pooling, deep penetration, aspiration), suggesting a possi- ment was 0.95 (95% CI 0.89–1.00) for postswallow pyri- ble underlying somatic cause, five visuoperceptual ordinal form sinus pooling. The lowest level of intraobserver variables (piecemeal deglutition, postswallow vallecular agreement was 0.90 (95% CI 0.80–1.00) for postswallow pooling, postswallow pyriform sinus pooling, laryngeal vallecular pooling. The prevalence of impairment was very penetration, and aspiration) were scored by two indepen- low for all variables. dent judges [13, 21–26]. All of these variables were scored for every FEES swallow at varying speed. The judges FEES Variables underwent consensus training for these measurements, as described previously [13, 21–26]. Both judges were blin- Descriptive data of the FEES variables are displayed in ded to the patients’ identity and medical history. The jud- Table 3. Piecemeal deglutition was rated as normal (cate- ges were also blinded to each other’s scores. To determine gory 0) in 31.0% (N = 13), 16.7% (N = 7), and 7.1% intraobserver agreement, 30 (29%) of the FEES swallows (N = 1) of the swallows for thin liquid consistency, thick were rated twice (repeated measurements). These FEES liquid consistency, and bite-sized cracker, respectively. In swallows were randomly selected and again blinded for five patients, postswallow vallecular pooling was rated as both judges. Fatigue-related observer bias was avoided by mild (14.3 and 7.1% of the swallows for thin liquid and limiting the judge’s rating task to two hours per session. thick liquid consistency, respectively), but in none of these patients was pooling observed in all seven recorded swal- Statistical Analysis lows. All five patients showed at least one normal swallow without vallecular pooling. None of the swallows was rated Levels of interobserver and intraobserver agreement were as severe vallecular pooling (category 2). Penetration was measured for each variable by the linear weighted kappa observed in two patients. The first patient showed a trace of 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Table 1 Patients’ characteristics Subject Age Gender BMI MMSE- Psychiatric history Psychiatric Referred by No. of visits score medication otorhinolaryngology outpatient clinic MUMC? 1 56 Female 29 30 – – GP 6 2 27 Male 17 30 Pervasive developmental – Internist 1 disorder—not otherwise specified 3 43 Male 17 25 Cluster B personality Temazepam, GP 2 disorder Oxazepam 4 41 Female 21 23 Panic disorder Citalopram Otorhinolaryngologist 3 5 51 Male 23 30 – – MV 1 6 68 Male 25 29 – – GP 1 7 26 Male MV 23 – – GP 9 8 53 Male MV 23 – – Otorhinolaryngologist 3 9 63 Female MV 26 – – GP 1 10 19 Female 16 23 – – GP 1 11 60 Female 37 29 Psychotic depression Quetiapine Neurologist 1 12 61 Female 34 29 – – Internist 1 13 34 Female 20 30 – – GP 2 14 66 Male 25 30 – – Pulmonologist 2 BMI Body Mass Index, MMSE mini mental state examination, GP general practitioner, MV missing value, MUMC? Maastricht University Medical Center methylene blue on the laryngeal side of the epiglottis DSS during the first thin liquid swallow. The second patient showed deeper penetration, near the vocal folds, in multi- The median score for the DSS was 66.0 (18–100). Spear- ple swallows and was therefore excluded because an man’s rho revealed no significant correlation between age underlying somatic cause of OD could not be excluded. and DSS. The DSS was not significantly different for None of the patients showed aspiration or pyriform sinus patients with clinically relevant symptoms of anxiety or pooling during the swallowing examination. The study depression compared to patients without symptoms of population was too small to perform further statistical anxiety or depression. Males scored significant higher on analyses. the DSS compared to females. See Table 4 for the results of the Mann–Whitney U tests for group comparison. Hads Discussion Six of the 14 participants (42.8%) showed clinically rel- evant symptoms of anxiety (score C 8 on the anxiety This is the first study that investigates swallowing function subscale). Three of the 14 (21.4%) showed clinically in relation to symptoms of anxiety and depression in relevant symptoms of depression (score C 8 on the patients with MUNOD. All 14 included patients presented depression subscale). These three also had a score C 8on with complaints of OD, and none showed structural the anxiety subscale. Thus, 42.8% (N = 6) of the partic- abnormalities during FEES examination. However, the ipants had clinically relevant symptoms of anxiety and/or majority showed abnormal piecemeal deglutition, which depression. The Chi-squared test showed no gender dif- could be an early symptom of an underlying somatic dis- ferences between patients with and without clinically order impairing normal swallowing physiology. However, relevant symptoms of anxiety (p = 0.28) or depression it is conceivable that abnormal piecemeal deglutition is a (p = 0.51). The Mann–Whitney U test showed no age clinically relevant symptom of MUNOD. Since these differences between patients with and without clinically patients are often anxious about swallowing, multiple relevant symptoms of anxiety (p = 1.00) or depression swallows of smaller fragments of the same bolus may offer (p = 0.76). them a sense of safety or control. In these patients, 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Table 2 Interobserver and intraobserver agreement levels per FEES variable assessed with linear weighted Kappa and 95% confidence interval FEES outcome Definition Ordinal scale Interobserver Intraobserver agreement (95% CI) variable agreement (95% Observer 1 Observer 2 CI) Piecemeal Sequential swallowing on the Five-point scale (0–4) 0.99 (0.97–1.00) 0.93 (0.84–1.00) 0.93 (0.84–1.00) deglutition same bolus 0 = no additional swallows 1 = one additional swallow 2 = two additional swallows 3 = three additional swallows 4 = four additional swallows Postswallow Pooling in valleculae after the Three-point scale (0–2) 0.95 (0.91–1.00) 0.96 (0.89–1.00) 0.90 (0.80–1.00) vallecular swallow 0 = no pooling pooling 1 = filling of less than 50% of the valleculae 2 = filling of more than 50% of the valleculae Postswallow Pooling in pyriform sinuses after Three-point scale (0–2) 0.95 (0.89–1.00) 1.00 1.00 pyriform sinus the swallow 0 = no pooling pooling 1 = trace to moderate pooling 2 = severe pooling up to complete filling of the sinuses Penetration and Penetration of bolus in the Three-point scale (0–2) 0.98 (0.96–1.00) 0.97 (0.90–1.00) 0.97 (0.90–1.00) laryngeal vestibule, above the aspiration 0 = no penetration vocal folds 1 = penetration Aspiration of bolus below the 2= aspiration vocal folds Results of intraobserver agreement are given for both observers Lower scores refer to normal functioning, whereas higher scores refer to more severe disability Kappa values: \ 0 = less than chance agreement; 1 = perfect agreement piecemeal deglutition seems to be a habitual coping strat- [28]. The bladder–gut–brain axis is an interesting frame- egy rather than a subclinical neurogenic impaired swal- work. It suggests a bidirectional pathway between brain lowing pattern. Nevertheless, follow-up for a possible and body, assuming that both functional and affective progressive neurologic disease is recommended. It is disorders are stress related and that functional symptoms assumed that swallowing physiology in patients with are a sensitized response to earlier threats. This sensitiza- MUNOD is normal. However, an interesting question is tion might mediate false-alarm signals (alarm falsification whether MUNOD could disturb normal swallowing phys- as a defense system). That, in turn, could provoke emo- iology. Roland et al. evaluated the incidence of esophageal tional and physical distress, resulting in psychiatric con- contractility disturbances in psychiatric patients [27]. ditions and functional disorders like MUNOD [6, 28]. A Manometry showed a high percentage of functional motor study by Dum et al. raised the possibility that motor areas impairment in patients with complaints of anxiety and/or of the cerebral cortex are important in the stress and depression, while endoscopy in these patients showed no depression connectome [29], and Grillon et al. suggested structural abnormalities [27]. In a large prospective popu- that anxiety increases motor response inhibition [30]. lation-based study, Koloski et al. showed that anxiety is an These studies indicate a relationship between affective independent predictor for new onset functional gastroin- function and motor function and thus strengthen the testinal disorders like irritable bowel syndrome, suggesting assumption that functional complaints might be part of a that affective disorders can underlie physical symptoms hypersensitivity or alarm-falsification disorder [6]. By 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia Table 3 Frequency distribution FEES category frequencies of swallows per category of the different FEES variables, given Thin liquid consistency Thick liquid consistency Bite-sized cracker as absolute numbers (N) and N (%) N (%) N (%) percentages (%) N =42 N =42 N =14 Piecemeal deglutition Category 0 13 (31.0) 7 (16.7) 1 (7.1) Category 1 10 (23.8) 15 (35.7) 2 (14.3) Category 2 13 (30.9) 10 (23.8) 2 (14.3) Category 3 1 (2.4) 3 (7.1) 2 (14.3) Category 4 5 (11.9) 6 (14.3) 6 (42.9) MV 0 1 (2.4) 1 (7.1) Postswallow vallecular pooling Category 0 35 (83.3) 36 (85.7) 12 (85.7) Category 1 6 (14.3) 3 (7.1) 0 Category 2 0 0 0 MV 1 (2.4) 3 (7.1) 2 (14.3) Postswallow pyriform sinus pooling Category 0 41 (97.6) 40 (95.2) 12 (85.7) Category 1 0 0 0 Category 2 0 0 0 MV 1(2.4) 2 (4.8) 2 (14.3) Penetration/aspiration Category 0 40 (95.2) 41 (97.6) 12 (85.7) Category 1 1 (2.4) 0 0 Category 2 0 0 0 MV 1 (2.4) 1 (2.4) 2 (14.3) Missing value; FEES variable could not be rated deglutition). So far, no studies have been published on this Table 4 Comparison of DSS between patients with clinically relevant symptoms of anxiety or depression and patients without symptoms of subject. However, the assumption that patients with anxiety or depression using Mann–Whitney U test and comparison of MUNOD must have a normal swallowing function might DSS between male and female patients using the Mann–Whitney be incorrect. Through this bidirectional pathway, a psy- U test chiatric problem can have sensorimotor effects on the N DSS score Level of significance swallowing function without there being any other cause of Median (range) p-value dysphagia, such as a chronic neurological disorder. Then, it HADS-D C 8 3 85.0 (18–100) 0.659 would be plausible that OD can be caused by affective HADS-D \ 8 11 57.0 (31–98) disorders or psychiatric conditions, even when the swal- HADS-A C 8 6 76.0 (18–100) 0.662 lowing physiology is disturbed. In this study, none of the HADS-A \ 8 8 55.5 (31–98) participants had symptoms indicating an underlying Male 7 85.0 (44–100) 0.017 somatic disease, and none showed other abnormalities Female 7 54.0 (18–77) during structured interviews or general otorhinolaryngol- ogy examination (normal cranial nerve integrity, speech, DSS Dysphagia Severity Score, HADS-D Depression subscale of the etc.). Although a somatic cause of dysphagia might seem Hospital Anxiety and Depression Scale, HADS-A Anxiety subscale of the Hospital Anxiety and Depression Scale unlikely, MUNOD should always be a diagnosis of Statistically significant exclusion. Previous research showed a high prevalence of clinically relevant affective symptoms in OD patients [13, 14, 31]. The present study underpins these data. It also shows a high implication, MUNOD and functional motor impairment prevalence (42.8%) of clinically relevant affective symp- may be interrelated too, causing disturbances of the normal toms, which indicates that MUNOD seems to be related to swallowing physiology (such as increased piecemeal 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia affective conditions in more than 40% of the cases. Four of prolonged dysphagic complaints, with no indication of a the participants (28.5%) had already been diagnosed with a somatic disease or abnormality, psychiatric conditions psychiatric condition (psychotic depression, panic disorder, must be considered as a possible cause of OD. Validated pervasive developmental disorder—not otherwise speci- psychological screening questionnaires could be helpful in fied, cluster B personality disorder). The patient with the detection of affective conditions but also of other cluster B personality disorder showed clinically relevant psychiatric conditions. Involvement of a psychiatrist and/or symptoms of anxiety and depression, and the patient with psychologist is recommended. panic disorder exhibited clinically relevant symptoms of anxiety. In these patients, MUNOD and affective symp- Limitations of the Study toms are likely to be part of their psychiatric disorder. The DSS scores were not significantly different between This investigation has some limitations. First, since patients with and without clinically relevant affective MUNOD is a rare condition, the number of patients symptoms. Apparently, clinically relevant symptoms of included in the study is small, so only a limited statistical anxiety and depression are not related to the severity of analysis could be performed. Second, the HADS ques- MUNOD symptoms. A psychological screening question- tionnaire was used for screening of anxiety and depression naire, like the HADS, is a simple tool for the preliminary symptoms. Possibly, a different screening tool or multiple assessment of the affective state of a patient. However, the screening tools would have led to different results. Third, expertise of a psychiatrist is essential to a definitive diag- three of the participants were taking psychiatric medication nosis and treatment of any psychiatric condition, including (see Table 1), which could have a negative effect on phagophobia or other anxiety disorders, and depression. It swallowing [32, 33]. Furthermore, the use of psychiatric might be helpful to draw upon the patient’s psychiatric medication could have led to an underestimation of the history and to involve his or her own psychiatrist when HADS scores. Furthermore, this investigation used a cross- preparing a multidisciplinary treatment strategy. Involve- sectional study design and was not intended as a therapy- ment of a psychiatrist would obviously be necessary. effect study; the effect of different treatment options could However, the patient must be willing to cooperate and be examined in future research, which could also specify accept that a psychiatric problem might be the cause of the treatment strategies in patients with MUNOD and psychi- swallowing problems. In this study, only four patients atric comorbidity. could be convinced to visit a psychiatrist after visiting the outpatient clinic for dysphagia. Following referral to the psychiatrist, one patient was diagnosed with an anxiety Conclusion disorder and one patient was diagnosed with an identity disorder. Two of the referred patients were already known MUNOD is a rare condition that is difficult to diagnose. with a psychiatric disorder (panic disorder and psychotic We hope to help dysphagia caregivers by sharing our depression), see Table 1. Early recognition of MUNOD results and experiences. Patients deserve a professional and a motivational trajectory towards integrated care are approach, particularly because their diagnostic trajectory necessary to develop effective treatment strategies, to has often been long and inconclusive. Affective symptoms reduce health care consumption and health care costs, to are common in these patients. MUNOD could be a symp- decrease the risk of iatrogenic damage arising from con- tom of a psychiatric condition or part of the alarm falsifi- tinuous diagnostic intervention, and to prevent frustration cation defense system, suggesting that physical symptoms in the interaction between physician and patient [11]. and affective disorders are stress-related and a response to Almost all of the participants had already consulted mul- earlier threats. Consultation of a psychiatrist for patients tiple specialists or had made recurrent visits to outpatient with MUNOD is recommended as part of a pathway toward clinics all over the Netherlands. Consultation of a psychi- multidisciplinary integrated care. atrist must be considered as an early option in the diag- nostic strategy of MUNOD instead of the ‘last resort’ after unsuccessful treatment. Diagnosis and treatment of an Compliance with Ethical Standards underlying psychiatric disease may improve the swallow- ing problems. It is important to realize that affective Conflicts of interest The authors declare that they have no conflict of symptoms are frequently present in patients with MUNOD. interest (financial or non-financial). Assuming a bidirectional pathway between brain and body, Ethical Approval The authors declare that this manuscript has not MUNOD could be understood as a symptom of physical been submitted to other journals and that this manuscript has not been distress or part of an alarm falsification and defense reac- published previously. Consent to submit was received from all co- tion as seen in other functional syndromes. In patients with 123 R.J.C.G. Verdonschot et al.: Medically Unexplained Oropharyngeal Dysphagia authors, and all co-authors contributed sufficiently to the scientific oropharyngeal dysphagia. J Psychosom Res. 2013;75(5):451–5. work. Informed consent was obtained from all patients included in https://doi.org/10.1016/j.jpsychores.2013.08.021. this study. 15. Folstein MF, Folstein SE, McHugh PR. ‘‘Mini-mental state’’. A practical method for grading the cognitive state of patients for the clinician. J Psychiatr Res. 1975;12(3):189–98. 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DysphagiaSpringer Journals

Published: Jun 5, 2018

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