Functional Voice Disorders: The Importance of the Psychologist in Clinical Voice Assessment

ARTICLE IN PRESS Functional Voice Disorders: The Importance of the Psychologist in Clinical Voice Assessment *Mafalda Andrea, †Óscar Dias, †Mário Andrea, and ‡Maria Luísa Figueira, *†‡Lisbon, Portugal Summary: Objectives. The etiopathogenesis of functional voice disorders (FVDs) is multifactorial. The purpose of this study was to analyze the severity of depression and anxiety, and the incidence of affective and anxiety disorders, in patients who presented different types of FVDs and were followed at the University Clinic of Otolaryngology. Design. This is a cross-sectional study. Methods. After ENT observation, 83 women were classified into three groups: psychogenic voice disorder (PVD = 39), primary muscle tension voice disorder (MTVD1 = 16), and secondary muscle tension voice disorder (MTVD2 = 28). A psychologist assessed the severity of depression and anxiety using the Hamilton rating scales, and screened for affective and anxiety disorders using the Mini International Neuropsychiatric Interview. Results. Significant differences in the mean values were found between the groups, with the MTVD1 group having higher levels of depression and anxiety. In affective disorders (current major depression and current mood disorder with psychotic symptoms) and in anxiety disorders (lifetime panic disorder, current generalized anxiety, and current panic disorder with agoraphobia), significant differences in association were found between groups. Conclusions. Groups presented with significant differences in depression and anxiety levels, and in some psychiatric diagnoses. Patients with FVDs should be independently assessed regarding their voice disorder classification. The integration of a psychologist in the clinical voice assessment team is essential, as findings have corroborated an important incidence of psychiatric disorders in FVDs patients. Key Words: Functional voice disorders–Psychogenic voice disorders–Primary muscle tension voice disorders–Secondary muscle tension voice disorders–Psychiatric Disorders. INTRODUCTION In the adult population, the prevalence rate of voice disorders was estimated at 7.6%,1 with a lifetime prevalence rate of nearly 30.0%.2,3 Vocal symptom presentation ranges in a continuum from the complete absence of voice (aphonia) to varying degrees of vocal impairment (dysphonia).4,5 The vocal symptom may also have a temporary or prolonged expression.1 A balance between active and attentive listening is needed when patients present to ENT specialists with voice symptoms.6 It is up to the voice professional to focus on voice quality7 and the patient’s subjective perception of his or her voice problem. The medical evaluation encompasses not only the observation of the laryngeal structures,8 but also the vocal folds particularly during breathing and phonation. Voice disorders are multidimensional and their classification contributes in the characterization of the etiology, the anatomofunctional presentations, and the biopsychosocial factors that they are associated with. The establishment of a classification system has implications on the definition of the treatment choice for each patient with voice disorders. Several approaches are used to classify the broad range of voice disorders,9 yet the most common classification corresponds to the dichotomous classification between organic and functional

Accepted for publication October 20, 2016. From the *Department of Psychiatry, Santa Maria University Hospital, Lisbon, Portugal; †University Clinic of Otolaryngology, Faculty of Medicine, University of Lisbon, Lisbon, Portugal; and the ‡University Clinic of Psychiatric and Medical Psychology, Faculty of Medicine, University of Lisbon, Lisbon, Portugal. Address correspondence and reprint requests to Mafalda Andrea, Department of Psychiatry, Santa Maria University Hospital, Avenida Professor Egas Moniz, 1649-035, Lisboa, Portugal. E-mail: [email protected] Journal of Voice, Vol. ■■, No. ■■, pp. ■■-■■ 0892-1997 © 2016 The Voice Foundation. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jvoice.2016.10.013

voice disorders (FVDs), with FVDs being classified as psychogenic voice disorders (PVDs), primary muscle tension voice disorders (MTVD1), or secondary muscle tension voice disorders (MTVD2). Organic voice disorders are related to malformations of the larynx, acute or chronic inflammations of the vocal folds, vocal fold paralysis, or benign and malignant tumors.10 FVDs are characterized by dysphonia in the presence of apparent normal vocal fold anatomy and movement. This type of voice disorder may originate from psychological and idiopathic causes.11 In FVDs the voice has poor quality, ie, there is no relationship between voice quality and laryngeal signs. The abuse (any vocal behavior that strains or injures the vocal folds, like excessive talking, throat clearing, coughing, smoking, or yelling) or misuse (improper voice usage such as speaking too loudly or at an abnormally high or low pitch) of the anatomic and physiological vocal apparatus causes the vocal behavior to be located at the center of FVDs. A poor vocal technique can lead to the development of compensatory laryngeal maneuvers12–14 as an attempt to maintain the previous vocal register, which is associated with muscle tension. This muscle tension during phonation may result from precipitating factors such as acute laryngitis, laryngeal trauma, allergies, prolonged voice rest, persistent cough, or psychological stress.15 These factors in combination with anxiety or depression,2 high neuroticism,16 or the presence of personality disorders may contribute to the development of FVDs.7,17 Patients with FVDs were described as having interpersonal sensitivity or estrangement and distrust of others,18–20 and as being very reactive to stressful life events.21 The anatomo-functional predispositions were also identified: small glottic proportions, occupational susceptibility, prolonged stress exposure, and laryngeal inflammatory processes.12 All these factors can interfere

ARTICLE IN PRESS 2 in the etiopathogenesis of FVDs, such as worsening the vocal condition or delaying the patient’s vocal recovery. FVDs are more predominant in women than in men (3:1) and are more frequent between the third and fifth decades of life.15 For Aronson, a PVD indicates the existence of one or more psychological disorders: affective (depression and/or anxiety disorders), conversion, and/or personality disorder, and all can interfere negatively with the phonation process.22 The term psychogenic clearly indicates that the primary process has a psychological origin,23 reinforcing the complex and dynamic interaction between vocal production and personality and emotional status.20 In 1983, Morrison and colleagues24 introduced the term muscle tension dysphonia to describe a clinical feature in which patients evidence normal vocal fold morphology and movement. However, extensive voice use can lead to laryngeal changes, which then alter voice quality.11 Muscle tension dysphonia may manifest itself in two ways: as a primary or secondary feature.25 Muscle tension voice disorders have various etiologies26: poor vocal technique, great vocal demands and psychological factors,27 inappropriate vocal behavior, gastroesophageal reflux, and psychological and personality factors.28,29 Behlau and colleagues12 emphasized the role of psychological and/or personality factors and vocal misuse or abuse that lead to compensation with an increase on the vocal fold tension. Because muscle tension often arises from the overactivity of the autonomic and voluntary nervous system when arousal or anxiety occurs, poorly regulated laryngeal muscle activity patterns may be present.30,31 Muscle tension voice disorders affect nearly 10.0–40.0% of patients in a voice clinic and are also more prevalent in middleaged women.32 MTVD1 occurs in the absence of organic vocal fold pathology and is associated with excessive, atypical, or abnormal laryngeal movements during phonation.24 In a laryngeal examination, the most common feature is a wide gap upon closure and medial and anteroposterior compression of the glottis with reduction of the vocal fold amplitude.33 MTVD2 has both behavioral and organic etiologies. This particular feature corresponds to the vocal pathology in which the presence of an underlying organic condition causes undesirable changes to the function of the vocal folds. Consequently, the excessive effort made during voice production whenever the speaker tries to maintain his or her normal pitch and volume in the structurally altered larynx4,24 causes trauma to the vocal folds,25 resulting in vocal nodules, polyps, or cysts. This means that in the presence of fold lesions, a new voice production pattern will arise based on the excessive muscle activity24 and a vicious circle can be installed. In reference studies with FVDs patients, the design usually includes a group of patients with FVDs, often with a higher participation of women than men, and a control group composed of healthy subjects, matched by sex and age. Willinger and collaborators34 assessed the severity of depressive symptoms of 61 patients with functional dysphonia, and they concluded that 33% of the patients showed clinically significant depressive symptoms, with a percentage far superior to that obtained by the healthy control group. The authors also

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used a standardized psychiatric interview that allowed them to diagnose 33.0% of FVDs patients with mood disorders and 20.0% of FVD patients with anxiety disorders, based on the Diagnostic and Statistical Manual of Mental Disorders (DSM-IV) criteria for Axis I.34 Kotby et al35 evaluated the severity of anxiety symptoms in a comparative study involving 100 patients diagnosed with nonorganic voice disorders and 50 normal individuals (control group). Elements of both genders were present in both groups. For the sum of mild, moderate, and severe categories of anxiety severity, subjects with non-organic voice disorders scored 43.0% and individuals of the control group reached a value of 6.0%. Whenever FVDs are investigated, reference to psychiatric illness always arises. The majority of the scientific articles published before 2005, which aimed to address the relationship between FVDs and psychopathology, enabled us to verify that expressions such as “psychological distress,”12,23 “psychological/ emotional conflict,”21,36,37 “a response to negative emotions in the context of stressful life events,”21 “emotional maladjustment,”1,21,36,38 and “mental health troubles”39 are often used. These labels were chosen to refer to the existence of psychological components acting as precipitants, maintaining and/or perpetuating factors. A review of these studies raised some methodological issues: small sample sizes,17,40,41 a study group of both genders with a non-homogeneous number of participants being compared with a healthy control group,23,38,40,41 and whenever the aim was to analyze the frequency of psychological variables in populations with voice disorders, too often these studies resorted to the use of a semi-structured interview and self-report psychological standardized tests.2,23,36,38,39,41,42 In addition to these methodological issues, the choice of nomenclature raised the question about the real meaning of these labels as they only provide information about the presence of psychological variables in the etiopathogenesis of FVDs. These labels are not informative in respect to the psychological elements they refer to. More recently, terms such as depression, anxiety, and personality disorders are commonly used in literature when referring to FVDs patients either at a syndromatic or nosological level according to the DSM-IV and the International Statistical Classification of Diseases and Related Health Problems (ICD)-10 criteria. At present, an overall classification does not exist, and this is justified by the lack of consensus on nomenclature. The theoretical model underlying this study recognizes the existence of organic and functional voice disorders, and we will classify FVDs as one of the three presentations that were previously described. Patients with FVDs were referred to as a group vulnerable to the development of psychiatric disorders. An exploratory and cross-sectional study was designed to assess psychiatric disorders in FVDs patients who were classified into three groups: PVD, MTVD1, and MTVD2. The main purpose of the present study was to explore and compare the severity of depression and anxiety within groups, which were composed solely of female FVDs patients, and to investigate and compare the incidence of affective and anxiety disorders between groups with the application of hetero assessment standardized tests.

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METHODS Ethics statement The present study was approved by the Ethics Committee of the Faculty of Medicine, University of Lisbon/Santa Maria University Hospital, Lisbon, Portugal. All subjects were voluntary participants who signed written and informed consent forms before the evaluation. The data collection was made solely by the principal author of the study, a clinical psychologist, so as to assure anonymity and confidentiality. Subjects All patients with vocal complaints went directly to the University Clinic of Otolaryngology/Santa Maria University Hospital to seek voice care. They were later referenced to participate in the present study by different multidisciplinary professionals of the voice team, namely the ENT specialists and/or the voice therapists. Women are more vulnerable to the development of FVDs than men because of their structural differences in laryngeal anatomy43 and because of hormone-mediated effects.44 Compared with men, women also react differently to voice problems42 and have a greater risk of suffering from mood and anxiety disorders.45 Therefore, in order to define a constant variable (gender), the participation was restricted to women. Besides the exclusive participation of women, the following were defined as exclusion criteria: being under age (a minor), European Portuguese not their first language, illiteracy, inadequate hearing status, pregnancy, puerperium, breast-feeding, transsexuality, neurologic disease, history of current cancer disease, organ transplant history, and malignant lesion of the larynx and/or in otolaryngological territories. A total of 83 female patients with FVDs, aged between 18 and 83 and with a mean age of 52.51 years (standard deviation = 14.27), were eligible for inclusion in this study. Experimental procedure Patients with FVDs were screened in social, biomedical, otolaryngology, and psychological dimensions. A standard evaluation protocol was applied in order to establish both an otolaryngologist and psychiatric diagnosis. Clinical data A semi-structured interview consisting of open and closed questions with yes or no answers was designed to explore the patient’s sociodemographic information, habits, past and present medical history, voice use patterns, voice symptoms and signs, current and/or past history of voice therapy treatment, family history of voice disorders, psychiatric medical history, current and/or past history of treatment in mental health services, and family history of psychiatric disorders. Overall, it allowed us to construct the medical history of each patient. Otolaryngology evaluation All patients were observed and evaluated by an ENT specialist using videoendoscopy through rigid endoscopy (Karl Storz en-

3 doscopes; Tuttlingen, Deutschland) or flexible laryngeal fibroscopy (Olympus fiberscope; Auckland, New Zeland) examinations, which permitted the recording of the vocal folds in digital format. The endoscopic findings were analyzed separately by two other ENT specialists who made their own diagnoses for each subject. If disagreement arose, the images were reanalyzed. Classification criteria. In the videoendoscopic records, the vocal folds were captured in the abduction and adduction positions, allowing the evaluation of their anatomy and movement. Lastly, the classification of the patients with FVDs was made by the voice team, having as reference the underlying theoretical model that was previously described. The patients were classified and clustered into three groups: PVD group with 39 patients (47.0%), MTVD1 group with 16 patients (19.0%), and MTVD2 group with 28 patients (34.0%). Psychological evaluation The battery of psychological tests included the following standardized tests: Hamilton Depression Rating Scale, Hamilton Anxiety Rating Scale, and Mini International Neuropsychiatric Interview. The Hamilton Depression Rating Scale (HAM-D) is a semistructured, clinician-rated interview that was developed by Max Hamilton in 1960, and is the most used tool to assess depression severity worldwide. The original scale contains 17 items, which are classified in categories according to the increase of intensity. Each item can be scored on a 5-point (absent, mild, moderate, severe or incapacitating) or 3-point scale (absent, slight or trivial, clearly present).46 The depression severity is reached by totaling the scores of the following categories: depressed mood, suicide, work and loss of interest, retardation, agitation, gastrointestinal symptoms, general somatic symptoms, hypochondriasis, insight, and weight loss.47 The sum of scores is classified into normal or remission (0–7), mild depression (8–13), moderate depression (14–18), severe depression (19–22), and very severe depression (≥23). This means that the higher the score, the more severe the depression (0–54).48 The psychometric properties reveal an excellent validation and adequate internal consistency of different versions (range from .48 to .92),46 and the inter-rater reliability of the scales is consistent and exceeds .85.48 In this study, the internal consistency coefficient of the HAM-D was good, with a Cronbach’s α of .82. The Hamilton Anxiety Rating Scale (HARS), developed by Max Hamilton in 1959, is a rating scale that was developed to assess and quantify the symptom severity49 of somatic anxiety (physical complaints related to anxiety, such as insomnia, somatic muscular, somatic sensory, cardiovascular symptoms, respiratory symptoms, gastrointestinal symptoms, genitourinary symptoms, and autonomic symptoms) and psychic anxiety (mental agitation and psychological distress like anxious mood, tension, fears, and depressed mood).50,51 It is composed of 14 items categorized according to a series of symptoms (psychological and somatic anxiety) that can be classified into a 0- to 5-point scale, ranging from not present (score of 0) to severe (score of 4). The total score ranges from 0 to 56 and is reached by the sum of scores of each item. There are cutoff scores that indicate normal

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range (0–13), mild (14–17), moderate (18–24), and severe (25– 30) anxiety categories.51 The HARS is also a well-validated tool with acceptable levels of inter-rater reliability and is easy for the clinician to apply.51 In this study, the internal consistency coefficient of the HARS was good, with Cronbach’s α of .87. The Mini International Neuropsychiatric Interview v.5.0.0 (MINI) is a brief standardized diagnostic interview in clinical practice and research.52 It was developed in 1990 by two psychiatrists, David V. Sheehan (United States) and Yves Lecrubier (France), for the diagnosis of psychiatric disorders described in the DSM-IV and in the ICD-10.53 The MINI’s structure is based on the criteria of the DSM-III-R and IV54 and was designed to explore the Axis I of DSM-IV. This interview is organized in independent diagnostic modules that confer a high level of sensitivity. Each diagnostic section has two to four screening questions52 with a dichotomous codification (yes or no), and it encompasses algorithms that allow the establishment or exclusion of a diagnosis.55 The MINI allows us to make the following diagnoses: major depression disorder (current or recurrent), major depression with melancholic features (current), dysthymia (current), suicidality (current), manic episode (current or past), hypomanic episode (current or past), panic disorder (current or lifetime), agoraphobia (current), social phobia (current), obsessive-compulsive disorder (current), posttraumatic stress disorder (current), alcohol dependence (past 12 months), alcohol abuse (past 12 months), substance dependence (non-alcohol in the past 12 months), substance abuse (non-alcohol in the past 12 months), psychotic disorder (lifetime or current), mood disorder with psychotic features (lifetime or current), anorexia nervosa (current), bulimia (current), generalized anxiety disorder (current), and antisocial personality disorder (optional) (lifetime).56 In this paper, we used only the diagnostics of depression and anxiety according to the DSM-IV and ICD-10 criteria. When reference is made to lifetime psychotic disorder, the data report the presence of delusions and hallucinations that were scored as bizarre in MINI. Statistical data analysis Data were analyzed with SPSS Statistic v21.0 for Mac OS (IBM Corporation, Chicago, IL). Descriptive statistics measures were used to characterize the three groups: means, standard deviations, and minimum and maximum values for

continuous variables, and frequencies, percentages, and adjusted residuals for categorical variables. One-way analysis of variance (ANOVA) was applied to compare the groups’ depression (HAM-D) and anxiety (HARS) severity, whereas Tukey’s test procedure was used for multiple comparisons between groups. The chi-square test of association was used to evaluate the existence of associations between groups and psychiatric diagnoses (MINI). The correlation between the HAM-D and the HARS was assessed with Pearson’s linear correlation coefficient. The significance level was set at 5%. RESULTS First, all patients were assessed by an ENT specialist for the otolaryngology diagnoses and then classified into one of the three groups of FVDs. A psychologist evaluated the depression and anxiety severity and assessed the psychiatric diagnoses with the application of psychological tests. Classification of the groups A total of 83 female patients with FVDs were included in this study and classified into three groups: PVD group (39 subjects), MTVD1 group (16 subjects), or MTVD2 group (28 subjects). Demographic characteristics of the groups Considering the two variables, age and years of education (Table 1), with regard to age there were no large variations of ages by group, and no significant differences were found between the means of the three groups using one-way ANOVA (F[2,80] = 1.377, P = 0.258). Years of education presented a variability that was similar among groups, and there were no statistically significant differences between the means of groups tested with one-way ANOVA (F[2,80] = 2.011, P = 0.141). For the marital status variable (Table 2), the chi-square test showed that there were significant differences in association between group and marital status (χ2 = 15.424, df = 6, P = 0.017). In the MTVD1 group, there were more single patients than expected, and there were no divorced or widowed patients, whereas in the PVD group there were fewer single patients and more divorced patients than expected.

TABLE 1. Descriptive Statistics: Mean, Standard Deviation, and Minimum and Maximum for Age and Years of Education by Group

Age

Years of education

PVD MTVD1 MTVD2 PVD MTVD1 MTVD2

Mean

SD

Minimum

Maximum

55.1 48.6 51.2 8.6 9.5 10.8

11.97 17.50 15.04 4.29 5.16 4.32

28 18 21 4 3 3

79 71 83 17 17 19

Abbreviations: MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder; SD, standard deviation.

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TABLE 2. Descriptive Measures: Frequency, Percentage, and Adjusted Residual for Marital Status by Group

PVD

Frequency (percentage) Adjusted residual Frequency (percentage) Adjusted residual Frequency (percentage) Adjusted residual

MTVD1 MTVD2

Single

Married

Divorced

Widowed

2 (5.1%) −2.1 6 (37.5%) 3.2 3 (10.7%) − .5

24 (61.5%) −.2 10 (62.5%) .0 18 (64.3%) .2

10 (25.6%) 2.0 0 (0.0%) −2.0 4 (14.3%) −.4

3 (7.7%) .2 0 (0.0%) −1.2 3 (10.7%) .9

Abbreviations: MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder.

Results of the psychological tests of the groups Table 3 presents the descriptive statistics for the HAM-D by group. The MTVD1 group had a higher mean than the other two groups and was also the group with greater minimum and maximum values. Taking into account the classification of depression severity proposed

TABLE 3. Descriptive Statistics: Frequency, Mean, Standard Deviation, and Minimum and Maximum for Hamilton Depression Rating Scale by Group

PVD MTVD1 MTVD2

n

Mean

SD

Minimum

Maximum

39 16 28

10.2 14.1 8.0

6.10 5.27 4.33

0 4 0

22 24 14

Abbreviations: MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder; SD, standard deviation.

by the author of the test, the mean value obtained by the MTVD1 group corresponded to moderate depression, whereas both PVD and MTVD2 groups received the classification of mild depression. Significant statistical differences between the means of the groups were found (F[2,80] = 6.469, P = 0.002, ηp2 = .139, medium effect). Post-hoc analyses using Tukey’s HSD test procedure revealed differences between PVD and MTVD1 groups (P = 0.040), and between MTVD1 and MTVD2 groups (P = 0.002) (Figure 1). Table 4 presents the descriptive statistics for the HARS by group. The MTVD1 group had a higher mean compared with the two other groups. The minimum values were the same for PVD and MTVD2 groups and slightly higher for the MTVD1 group, yet maximum value was reached in the PVD group. According to the classification of anxiety severity proposed by the author of the test, the mean value obtained by the MTVD1 group was classified as mild anxiety, whereas both PVD and MTVD2 groups received the classification “normal range.” Statistically significant differences in means between groups were found using one-way ANOVA (F[2,80] = 7.752, P = 0.001, ηp2 = .162, medium effect). The

FIGURE 1. Means of HAM-D by group. HAM-D, Hamilton Depression Rating Scale; MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder.

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TABLE 4. Descriptive Statistics: Frequency, Mean, Standard Deviation, and Minimum and Maximum for Hamilton Anxiety Rating Scale by Group

PVD MTVD1 MTVD2

n

Mean

SD

Minimum

Maximum

39 16 28

11.6 16.3 9.6

6.36 4.79 4.20

1 7 1

29 21 18

Abbreviations: MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder; SD, standard deviation.

differences between PVD and MTVD1 groups (P = 0.013) and between MTVD1 and MTVD2 groups (P = 0.001) were reported by Tukey’s HSD test (Figure 2). To evaluate the relation between the HAM-D and the HARS, Pearson’s correlation coefficient was applied and a strong linear correlation (r = .877, P < 0.001) was found. This means that patients with a high level of depression severity also presented higher anxiety severity. All patients were screened with the MINI, which permitted psychiatric diagnoses. The chi-square test was used to assess the existence of associations between group and psychiatric diagnoses. There were statistically significant associations between group in five diagnoses that were classified in the two major diagnostic categories (affective and anxiety disorders), in lifetime psychotic disorder and in suicidiality. Table 5 presents the cross tabulations, group versus affective disorders (MINI), with two diagnoses evidencing significant associations, ie, current major depression (χ2 = 5.999, df = 2, P = 0.050) and current mood disorder with psychotic symptoms (χ2 = 6.202, df = 2, P = 0.045). The MTVD1 group reached a higher

TABLE 5. Descriptive Measures: Frequency, Percentage, and Adjusted Residual for Statistically Significant Affective Disorders in MINI for Each Group Affective Disorders

No

Yes

Current major depression PVD

Frequency (percentage) Adjusted residual MTVD1 Frequency (percentage) Adjusted residual MTVD2 Frequency (percentage) Adjusted residual

22 (56.4) 17 (43.6) −.9 .9 7 (43.8) 9 (56.2) −1.6 1.6 22 (78.6) 6 (21.4) 2.3

−2.3

Current mood disorder with psychotic symptoms PVD

Frequency (percentage) Adjusted residual MTVD1 Frequency (percentage) Adjusted residual MTVD2 Frequency (percentage) Adjusted residual

29 (74.4) 10 (25.6) −.8 .8 10 (62.5) 6 (37.5) −1.7 1.7 26 (92.9) 2 (7.1) 2.3 −2.3

Abbreviations: MINI, Mini International Neuropsychiatric Interview; MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder.

percentage compared with the other two groups, and in the MTVD2 group the analysis of adjusted residual indicates that there were fewer patients with current major depression in this group than expected. In current mood disorder with psychotic symptoms, the MTVD1 group reached the highest percentage, yet in the MTVD2 fewer patients than expected received this diagnosis.

FIGURE 2. Means of HARS by group. HARS, Hamilton Anxiety Rating Scale; MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder.

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TABLE 6. Descriptive Measures: Frequency, Percentage, and Adjusted Residual for Statistically Significant Anxiety Disorders in MINI for Each Group Anxiety Disorders

No

TABLE 7. Descriptive Measures: Frequency, Percentage, and Adjusted Residual for Statistically Significant Lifetime Psychotic Disorder Symptoms in MINI for Each Group

Yes

Lifetime panic disorder PVD

Frequency (percentage) Adjusted residual MTVD1 Frequency (percentage) Adjusted residual MTVD2 Frequency (percentage) Adjusted residual

33 (84.6) 2.4 7 (43.8) −2.8 20 (71.4)

6 (15.4) −2.4 9 (56.2) 2.8 8 (28.6)

−.1

.1

Current generalized anxiety PVD

Frequency (percentage) Adjusted residual MTVD1 Frequency (percentage) Adjusted residual MTVD2 Frequency (percentage) Adjusted residual

17 (43.6) 22 (56.4) −1.0 1.0 5 (31.2) 11 (68.8) −1.6 1.6 19 (67.9) 9 (32.1) 2.4

−2.4

Current panic disorder with agoraphobia PVD

Frequency (percentage) Adjusted residual MTVD1 Frequency (percentage) Adjusted residual MTVD2 Frequency (percentage) Adjusted residual

38 (97.4) 1 (2.6) .9 −.9 13 (81.2) 3 (18.8) −2.9 2.9 28 (100.0) 0 (0.0) 1.5 −1.5

Abbreviations: MINI, Mini International Neuropsychiatric Interview; MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder.

Table 6 presents the cross tabulations, group versus anxiety disorders (MINI), with emphasis on the three diagnoses where statistically significant associations were found, ie, lifetime panic disorder (χ2 = 9.474, df = 2, P = 0.009), current generalized anxiety (χ2 = 6.451, df = 2, P = 0.040), and current panic disorder with agoraphobia (χ2 = 8.619, df = 2, P = 0.013). Almost 28.0% of the patients had lifetime panic disorder, and the examination of adjusted residues showed that the PVD group had a lower percentage of patients with this diagnoses than expected, whereas in the MTVD1 group more patients received this diagnoses than expected. Half of the sample was diagnosed with current generalized anxiety, and the analysis of adjusted residual revealed that in the MTVD2 group there were fewer patients than expected with this diagnosis. Only 4.8% of the subjects were diagnosed with current panic disorder with agoraphobia, and through the analysis of the adjusted residual it was noted that in the MTVD1 group there were more patients receiving this diagnosis than expected. Table 7 presents the cross tabulations, group versus lifetime psychotic disorder category, by group (χ2 = 6.933, df = 2, P = 0.031). About 37.3% of the total of the sample were diagnosed as having symptoms related to a lifetime psychotic disorder, and the analysis of adjusted residuals revealed that there were fewer patients in the MTVD2 group with this diagnosis than expected. The MINI also showed significant statistical associations between group and suicidality (χ2 = 6.384, df = 2, P = 0.041).

PVD MTVD1 MTVD2

Lifetime Psychotic Disorder Symptoms

No

Yes

Frequency (percentage) Adjusted residual Frequency (percentage) Adjusted residual Frequency (percentage) Adjusted residual

21 (53.8) −1.6 8 (50.0) −1.2 23 (82.1) 2.6

18 (46.2) 1.6 8 (50.0) 1.2 5 (17.9) −2.6

Abbreviations: MINI, Mini International Neuropsychiatric Interview; MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder.

The suicide risk was more prevalent in the PVD group, either at the level of incidence or at the level of severity (Figure 3). DISCUSSION The present study was developed to analyze the severity of depression and anxiety, and to characterize the incidence of affective and anxiety disorders in three different groups of patients with FVD. In the literature, patients with FVDs were defined as a single study group that was subsequently compared with healthy controls. References to studies comparing the incidence of depression and anxiety in different types of FVDs were not found. Furthermore, in some studies, self-report instruments are often used to evaluate symptoms of depression and anxiety,2,23,36,39,42 making it impossible to compare the results derived from selfassessment standardized tests that report the patient’s perception of his or her symptoms, with results derived from clinical standardized scales that reflect the evaluation made by the mental health professional. A comparison could be made between our findings and the results of the Willinger and collaborators’ study.34 In fact, both researches administered the same assessment test for the evaluation of depression severity (HAM-D). In the Willinger and collaborators’ study,34 patients with functional dysphonia achieved a mean value of 13.3 in the HAM-D, thus receiving the classification of mild depression, whereas in our study the mean of the total sample was 14.1 corresponding also to the classification of mild depression. When the means for the three groups were analyzed, it was verified that they presented distinct values. The statistical analysis showed that the mean of the MTVD1 group was significantly higher than the mean of the other groups. Whereas the mean value obtained by the MTVD1 group corresponded to moderate depression, both the PVD and MTVD2 groups received the classification of mild depression, just like the study group in Willinger and collaborator’s research.34 In our study, 34.9% of the total of participants scored in mild, moderate or severe categories of the HARS. This result is similar to that of the Kotby et al. study35, which obtained a score of 43.0% for patients with no organic vocal disorders although it included participants of both genders. When our three groups were

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FIGURE 3. Frequency on the suicide risk categories by group. MTVD1, primary muscle tension voice disorder; MTVD2, secondary muscle tension voice disorder; PVD, psychogenic voice disorder. independently considered, percentages varied widely (MTVD1 = 75.0%, PVD = 38.5%, MTVD2 = 7.1%). This means that the classification of different groups considering the different types of FVD patients had a great impact on the anxiety analysis. The MTVD1 group stood out from the rest of the groups. The differences found between PVD and MTVD1 groups regarding the severity of depression and anxiety symptoms could be explained by the coping and defense mechanisms that were adopted by the patients of each group. The PVD patients seem to present a more exuberant clinical condition due to a mismatch between the signals (the functioning of the vocal folds) and the symptoms (the voice quality). Therefore, they are expected to be easily identified as having a mental illness, implying a faster referral to a proper treatment. In the future, more research on how patients with FVDs experience and express their inner suffering should be developed. A correlation between the HAM-D and the HARS was a novelty in studies with patients having voice disorders, but it is very important as it allows us to verify the existence of a strong correlation between depression and anxiety in FVDs. According to a systematic review, done between 1994 and 2009 on patients with chronic diseases, high rates of anxiety and depression are associated with increasing mortality rates and decreasing quality of life levels.57,58 To address this issue, the health system should promote early intervention in FVDs patients in order to prevent the development or aggravation of psychiatric disorders such as affective and anxiety disorders, and also to reduce or eliminate vocal symptoms. Therefore, the intervention of a mental health professional in the clinical voice assessment team is vital in order to provide an additional dimension to the care and comfort of these patients.59 The MINI enabled us to observe the existence of differences in association between groups and specific diagnoses of affective and

anxiety disorders and in the lifetime psychotic disorder category, with the MTVD1 group reaching higher percentages in all. We found a strong association between the risk of suicide and being in the PVD group. Regardless of the differences in risk of suicide observed between groups, this outcome deserves due prominence in future research and in the clinical context. In addition, despite the MINI not contemplating a diagnostic category for psychoses, it provided information about lifetime psychotic disorder symptoms, which presented significant differences between groups. According to Baker,21 there are virtually no reported cases of patients with FVDs suffering from psychotic disorders, yet in the MTVD1 and PVD groups about half of the participants presented lifetime psychotic symptoms. Future studies should take into consideration the incidence of psychotic disorders in patients with FVDs. In the MTVD1 group, an especially high prevalence of affective and anxiety disorders was identified. Future studies should explore more in depth the incidence of psychotic disorders in patients with FVDs. The cross-sectional design of the present study proved to be reliable for the evaluation of the incidence of affective and anxiety disorders, but it could not show causation between the psychological variables and FVDs. Our findings emphasize the need for future longitudinal studies of affective and anxiety disorders as causes or consequences of FVDs, particularly taking into consideration the classification of the three types of FVDs. For each FVDs presentation, it would also be important to identify the risk factors associated with its development to set a psychological profile and to recognize either bad or good prognosis factors connected to the treatment of the voice disorder or the psychoemotional state. The application of hetero assessment standardized psychological tests was fundamental to this study as it provided access to a common language based on the DSM criteria.

ARTICLE IN PRESS Mafalda Andrea, et al

Functional Voice Disorders

CONCLUSION Our findings provided evidence for a strong link between affective and anxiety disorders and FVDs. These FVDs patients presented high levels of comorbidity, proving the need for both clinical voice assessment and mental health care. The three groups—PVD, MTVD1, and MTVD2—received exactly the same kind of approach, which allowed us to see that even though they belong to the same category of the voice disorder classification, they do not present the same psychiatric diagnosis or the same degree of severity of depression and anxiety symptoms. As verified, whenever we explore severity of anxiety, we should take into account the type of FVDs presented by the patients. This indicates that their differences go beyond anatomo-functional presentations and voice quality, thus reinforcing the need for a specific assessment for each FVDs presentation. This approach demonstrates the need for the psychologist to be integrated into the clinical voice assessment team from the beginning so as to complement the medical and voice therapist evaluations. The role of the psychologist is complex. In the psychological evaluation process, it is up to the psychologist to identify, understand, and manage the psychological issues on which the vocal symptom is based or linked to. REFERENCES 1. Byeon H. Prevalence of perceived dysphonia and its correlation with the prevalence of clinically diagnosed laryngeal disorders: the Korea National Health and Nutrition Examination Surveys 2010–2012. Ann Otol Rhinol Laryngol. 2015;124:770–776. 2. Misono S, Peterson C, Meredith L, et al. Psychosocial distress in patients presenting voice concerns. J Voice. 2014;28:753–761. 3. Pernambuco L, Espelt A, Balata P, et al. Prevalence of voice disorders in the elderly: a systematic review of population-based studies. Eur Arch Otorhinolaryngol. 2015;272:2601–2609. 4. Green M, Mathieson L. The Voice & Its Disorders. 6th ed. Philadelphia, PA: Whurr Publishers; 2009. 5. Butcher P. Psychological process in psychogenic voice disorder. Eur J Disord Commun. 1995;30:457–474. 6. Colton R, Casper J, Leonard R. Understanding Voice Problems: A physiological perspective for diagnosis and treatment. Baltimore: Lippincott Williams & Wilkins; 2006. 7. Scott S, Deary IJ, Mackenzie K, et al. Functional dysphonia: a role for psychologists? Psychol Health Med. 1997;2:169–180. 8. Cohen SM, Thomas S, Roy N, et al. Frequency and factors associated with use of videolaryngostroboscopy in voice disorder assessment. Laryngoscope. 2014;124:2118–2124. 9. Sapienza C, Ruddy B. Voice Disorders. San Diego, CA: Plural Publishing; 2009. 10. Seifert E, Kollbrunner J. An update in thinking about nonorganic voice disorders. Arch Otolaryngol Head Neck Surg. 2006;132:1128–1132. 11. Rosen C, Murry T. Nomenclature of voice disorders and vocal pathology. Otolaryngol Clin North Am. 2000;33:1035–1045. 12. Behlau M, Madazio G, Oliveira G. Functional dysphonia: strategies to improve patient outcomes. Patient Relat Outcome Meas. 2015;6: 243–253. 13. Koufman J, Blalock D. Functional voice disorders. In: Koufman J, Isaacson G, eds. The Otolaryngologic Clinics of North America, Vol. 24, 2. Philadelphia, PA: W. B. Saunders Company; 1991:1059–1073. 14. Koufman J, Isaacson G. The spectrum of vocal dysfunction. In: Koufman J, Isaacson G, eds. The Otolaryngologic Clinics of North America, Vol. 24, 5. Philadelphia, PA: W. B. Saunders Company; 1991:985–988. 15. Morente J, Izquierdo A. Transtornos de la voz: Del Diagnóstico al Tratamiento. Málaga, Spain: Ediciones Aljibe; 2009.

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