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Defining the psychiatric role in spastic dysphonia
Defining the PsychiatricRole in SpasticDysphonia Barry I. Ginsberg, M.D. Division of Liaison Psychiatry,
Mt. Sinai Medical Center, New York
Joel J. Wallack, M.D. Division of Consultation-Liaison
Psychiatry,
Beth lsrael Medical Center, New York
James J. Srain, M.D. Division of Liaison Psychiatry,
Mt. Sinai Medical Center, New York
Hugh F. Biller, M.D. Department
of Otolaryngology,
Mt. Sinai Medical Center, New York
Abstract: The authors evaluated 11 surgically-treated patients with spastic dysphonia, a phonation disorder of unclear etiology. The results indicate that the illness does not appear to be a somatoform disorder, but that stress may play a role in its expression, and that there may be secondary depression and anxiety. The experience of spastic dysphonics suggests that psychiatric treatments may be inappropriately applied to an illness without clear organic etiology, whereas, conversely, a proper psychiatric role may be rejected when effective medical or surgical treatment is available. The authors recommend that psychiatrists evaluating patients with illnesses of unclear etiology should be cautious in making a primary psychiatric diagnosis unless DSM-111 criteria are met.
Spastic dysphonia is a phonation disorder of unclear etiology that has not, to our knowledge, been previously reported in the psychiatric literature. Nevertheless, psychotherapy and somatic psychiatric therapies are often among the multitude of treatments these patients receive. The results of psychiatric treatment have been disappointing [l], and the role of psychiatrists in the treatment of these patients has come under attack [2]. This communication reports the results of a study of 11 surgically-treated spastic dysphonics, and seeks to redefine the proper role for psychiatrists in the overall care of patients with this disorder. Spastic dysphonia was first described by Traube in 1871 [3]. Berendes [4] described it as “a disorder of phonation which is characterized by a strained, 132 ISSN 0163~8343/88/$3.50
creaking, and choked vocal attack, a tense and squeezed voice sound that is accompanied by extreme tension of the entire phonatory system,” and likened it to “stuttering with vocal cords.” A recent profile of 200 cases 151, as well as earlier clinical descriptions, indicate that the disorder usually (but not always) begins in middle age, is somewhat more prevalent in women, has a gradual onset, and has a chronic course, remaining without significant change once it has reached a plateau. Although a Medline search was unable to find mention of spastic dysphonia in the psychiatric literature, the otolaryngologic literature has included psychiatric opinions and assumptions. Brodnitz [l] described spastic dysphonia as a conversion disorder “at the opposite end (from hysterical aphonia) of functional voice disorders.” Evidence cited in favor of this etiology includes the recovery of normal phonation while singing and when drugged with sodium amytal or alcohol, as well as the presence in some cases of emotional trauma or stress seemingly related to the onset of symptoms. Congruent with this point of view, psychotherapy and/or speech therapy were thought to be the treatments of choice, although reports of success were lacking. Moreover, Aronson and colleagues [6] had reported in 1968 that MMPI profiles of female spastic dysphonics showed elevation on the depression scale more so than on the hypochondriasis or hysteria scale. This contrasts with General HospitalPsychiatry 10,
132-137,
1988
0 1988 Elsevier Science Publishing Co., Inc. 52 Vanderbilt Ave., New York, NY 10017
Psychiatric Role in Spastic Dysphonia
“conversion V” seen in somatization: the hypochondriasis and hysteria scales are more elevated than is the depression scale. Psychiatric interviews in this study found that spastic dysphonia patients were rigid, conscientious, highly responsible people, but these traits did not differentiate them from other medically ill patients. The interviews generally failed to find distinct precipitating emotional conflict or trauma. Nonetheless, the view of spastic dysphonia as a psychoneurotic symptom predominated until 1976, when a surgical treatment was reported by Dedo [2]. Dedo performed a unilateral section of the recurrent laryngeal nerve (RLN), which paralyzes the vocal cord in the paramedian position, allowing a somewhat breathy, but usually improved, phonation. Subsequently, Dedo [7] reported highly positive results with the procedure on 212 patients affected by spastic dysphonia; other groups [8,9] have reported more mixed but generally positive results. One of the authors (HFB) [lo] described a nerve crush procedure in 1977; however, it was subsequently reported [ll] that the initially favorable results of the procedure did not last, and it was abandoned in favor of the RLN section. Concomitant with these reports, various organic etiologies have been proposed. Dedo [12] has suggested that a virally induced proprioceptive deficit is responsible. Other investigators [13,14] have favored central cranial nerve dysfunction. Since the advent of the surgical appproach, there has been a pendulum swing away from any appreciation of emotional factors in spastic dysphonia, and away from psychiatric involvement in the care of these patients. Dedo [2] reported that the belief in a psychoneurotic etiology “just causes time and money to be wasted on psychotherapy, at least for the vocal disorder itself, and it increases the patient’s guilt and emotional stress.”
Methods We attempted to contact each of the 26 patients who had been treated for spastic dysphonia with unilateral RLN crush and/or section on the otolaryngology service at Mt. Sinai Hospital from 1976 to 1984. Of these patients, 11 agreed to participate; 10 were interviewed in person, and 1, who was located out of state, by telephone. A semistructured interview was used; the patient was encouraged to trace in detail the history of the illness and the treatment received. The interview also included questions about the effect of stress on the vocal
Table 1. Treatment Patient First M.D. Total M.D.s Speech therapy Psychotherapy Hypnosis Biofeedback Acupuncture Psychiatric drugs Chiropractor Meditation/relaxation Psychotherapy Help voice? Psychiatric drugs help voice?
1234567891011 0” 0 PbO F’F F F 4 6 5 4 7 6 5 5 +++++ +++ + +++++ ++ + + + + ++++++ + +
F F F 10 3 10 + + +
NdNNNNN
N
Y’NNNNN
NN
++ +
+
“0, otolaryngologist. ‘I’, psychiatrist. ‘F, family doctor. “N, no.
‘Y, yes.
symptoms, medical and psychiatric history with particular attention to somatoform disorders, and the effect of the illness on work and social life. The subjects included 7 females and 4 males, whose age ranged from 34 to 63 with a mean of 46. The age at the time of onset of symptoms ranged from 29 to 51, with a mean of 36. One patient had completed only high school, three had completed at least 2 years of college, five had been graduated from college, and two had graduate degrees. Eleven were white and one was Hispanic. Eight were married, one divorced, one widowed, and one had never married.
Results The experience of the patients in obtaining treatment (summarized in Table 1) indicates a pattern of widespread difficulty in diagnosis and treatment. Seven patients originally presented to family physicians, three to otolaryngologists, and one to a psychiatrist. Each patient subsequently saw at least three other specialists with a mean of 5.5; patients received a mean of 3.6 different kinds of treatment. Seven patients saw psychiatrists, with one of these patients also seeing a psychologist and a pastoral counselor. They each received psychotherapy as well as psychopharmacotherapy (sometimes from a physician other than the psychiatrist); one other patient who did not see a psychiatrist 133
B. I.
Ginsberg et al.
Table 2. Patients’ evaluation of surgery
Patient RLN crush RLN section
1
2
3
4
5
6
7
8
(-)
(+)
(-)
(+I--)
(-)
(+)
also received psychiatric medication. None of the patients felt that the psychotherapy was helpful for their speech disorder, although the patients frequently commented that their psychotherapy had been helpful in other ways. Seven of the eight patients who received psychiatric medication felt that it had not helped their speech, but one patient reported some improvement with alprazolam. Two of the patients had received a nerve crush procedure, one a crush followed by a section, and the other eight, a nerve section. Subjective evaluations of the effect of surgery upon the voice disorder is summarized in Table 2; all three who had had the crush felt that any benefit was only temporary and, overall, the procedure had not been helpful, the one who subsequently had the section, however, felt positively about that procedure. Of the eight patients who received only the section, four rated it positively, one was equivocal, and three were negative. This is similar to the followup results reported by Aronson and DeSanto [9] in a study of 33 patients, but is less positive than the results reported by Dedo and Izdebski [7], who studied 306 patients, and by Barton [8], who reported on 11 patients. Information germane to psychiatric diagnosis is contained in Table 3. Almost all the subjects reported a major stress (change of job, marital situation, financial situation, illness or death of someone close, major illness, or emotional problems) within the year prior to the onset of vocal symptoms. Most of the subjects reported that once the illness had begun, stress made the symptoms worse. Although four patients had had psychiatric treatment prior to their illness and the same number had psychiatric illness in the family, only one had a previous history of physical symptoms of unclear organic etiology, and none had a history of multiple medical illnesses; indeed, except for a not infrequent history of postnasal drip and other mild respiratory symptoms, the patients described themselves as usually healthy. None of the subjects had personally experienced similar vocal symptoms or had witnessed it in significant figures in their lives; such a “symptom model,” although not a criteron for conversion disorder in DSM-III 1151, 134
9 (-)
10
11
(+)
(+)
has been associated with that diagnosis [16]. Eight subjects reported significant depression (frequently including suicidal thoughts) secondary to the speech disorder, and two reported significant anxiety (usually about being called upon to speak) secondary to the disorder. Although six patients felt that their work status had been adversely affected by the illness and three felt that the illness had adversely affected their general social relations, none of the patients reported on adverse effect on marital relations.
Case Reports Patient 1 was a widow in her early fifties. She had been in psychotherapy since the death of her mother 4 years earlier, when she noted a “strain” and “tension” in her voice that she reported to have occurred after an upsetting session with her psychotherapist. She and her therapist had begun to discuss the possibility of terminating treatment and she was also involved at that time in leaving her home town. Concerns about financial independence were present at that time as well. During the next 6 months, the symptoms gradually worsened; she saw an otolaryngologist who referred her for speech therapy with little result. After the failure of speech therapy, she and her psychiatrist decided to intensively explore the possibility of a psychologic cause, and she resumed psychotherapy, which had been terminated as planned. Regardless of what she felt was progress in understanding, there was no effect on the vocal symptoms. She saw a second otolaryngologist, had another course of speech therapy, saw another psychiatristhypnotherapist (who, she reports, was sure that the vocal problem had a psychologic basis) for hypnosis, all without significant help. At periods of stress she took thioridazine and later trifluoperazine, without effect on the vocal symptoms; she did find that alprazolam provided some improvement for a period of hours. She received a nerve crush 3 years after the onset of symptoms with relief lasting about 6 weeks, then a nerve section 26 months later, resulting in an almost normal voice for the next 2 years until the return of some milder symptoms 6 months prior to the study. There was no history of multiple somatic problems, nor of a symptom model. Patient 2 was in her mid-thirties when symptoms began. She had had a major disappointment in her
Psychiatric
Table 3. Psychiatric
diagnostic
Role in Spastic Dysphonia
information
Patient Major stress <1 yr prior to S.D.” Stress makes voice worse Past somatic ills and/or hypochondria Symptom model Past psychiatric history Family psychiatric history Depression/anxiety 2” to S.D. Work problems 2” to S.D. Marital problems 2” to S.D. Social problems 2” to S.D.
1
2
3
+”
+ +
+
+ + +
+ + +
+
+ +
4
5
6
7
8
9
+
+ +
+ +
+ +
+ +I-’
+ +
+
-t +
-t
+
+
+
+ +
10
+ +
11 + +
+ +
+
+
+
+
+
+
“S.D., spastic dysphonia. ‘C , present. ’ + i - , equivocal.
career and felt dissatisfied with her life, which prompted her to begin psychotherapy 3 months prior to the onset of symptoms. After she was told by an otolaryngologist that she had a normal larynx, she assumed that the voice disorder was an “interesting“ symptom related to the expression of feelings in the new psychotherapy and would disappear spolitaneously. The symptoms did not remit, however, and a stormy psychotherapy was terminated at the suggestion of her psychiatrist after 1 year. She later underwent group psychotherapy, pastoral counseling, two courses of speech therapy, hypnosis, and treatment with diazepam, all without significant effect on her voice disorder. She received a nerve crush some 20 years after the onset of symptoms, with improvement in phonation. However, she disliked the breathy quality to her speech and declined to have nerve section after the symptoms recurred. Patient 3, a successful professional, had the onset of symptoms in his late 20s. Although he could identify no specific environmental or emotional precipitant, he considered himself a “hyper” individual, and the first physician he contacted was a psychiatrist, “to eliminate that possibility.” He reported that the psychiatrist was never sure of the etiology of the speech disorder, but he continued in treatment irregularly during the next 3 years and was treated at various times with diazepam, oxazepam, lithium, and antidepressants, as well as with biofeedback. These treatments were not helpful for the speech disorder. He had three surgical procedures: a recurrent laryngeal nerve crush, with improvement in speech lasting 23 months; a recurrent laryngeal nerve section, with improvement lasting 6-7 months, and a superior laryngeal nerve section, without improvement. Patient 4 was 29 years old when she noticed a “hesitancy” in her voice that progressed into the clinical picture of spastic dysphonia. She reported that the otolaryngologist she saw several months later in-
dicated that she had either multiple sclerosis, myasthenia gravis, or a psychologic problem. She had an inpatient neurologic workup, including a placebo trial (with some relief of symptoms), leading to a referral to a psychiatrist. She began psychiatric treatment that lasted 3 years and uncovered a possible precipitant. About a year and a half before her symptoms began, an event had taken place about which the patient appeared to have had much unconscious guilt. Despite exploration of her guilty feelings, the voice showed no improvement, although she found that her newly gained insights were valuable. At one point, the psychiatrist referred her to another otolaryngologist, who reportedly told her that the problem was psychologic and sent her back to the psychiatrist. She also received speech therapy for 1 year without significant benefit, and was given diazepam, also unsuccessfully. She became depressed, stopped socializing, was hypersomnolent, and became somewhat obese. She received a nerve crush 3 years after the onset of the disorder, with temporary improvement, followed by a nerve section, with more lasting effect, although the voice problem remains at a less severe level.
Discussion Almost invariably, these patients had seen several doctors and received various treatments with poor results prior to surgery. This is consistent with other reports [2,5]; in fact, the first patient to receive a RLN section had seen 29 doctors over 17 years [2]. Most were told that the problem was psychogenic or, on their own, they felt it was psychogenic at some point during the course of the illness. Most received psychiatric treatment, consistently without benefit in terms of the speech
135
B. I. Ginsberg et al.
disorder. Yet, 9 of the 11 patients noted an association of stressful events with the onset of the symptoms, and this may help explain the frequency and persistence of psychiatric treatment in the face of such poor results. As the case reports illustrate, a highly suggestive psychologic precipitant is sometimes presented. Nonetheless, several features of the illness are inconsistent with conversion disorder. The illness usually has a gradual onset and continues uninterrupted for many years; DSM-III notes that conversion disorder “probably is usually of short duration, with abrupt onset and resolution.” In all 11 patients, no symptom model was present. The previously cited study by Aronson and associates [6] found that MMPI and psychiatric interviews did not indicate that these patients tend toward somatization, and the histories of the patients in the present study were inconsistent with a diagnosis of somatization disorder. The interview data, albeit retrospectively, indicate that most patients with this disorder suffer anxiety and depression secondary to the vocal symptoms, and that stress seems to exacerbate the symptoms. The study data suggest that the most appropriate psychiatric diagnosis for most of these patients would be psychologic factors affecting physical condition (DSM-III code 316.00), although during the course of the illness, particularly prior to surgical treatment, some may have warranted a diagnosis of an adjustment disorder or, more rarely, major depression or an anxiety disorder. Although it is possible that further psychiatric exploration with these patients might have led to improvement of the vocal symptoms, the absence of any report of psychotherapeutic success with spastic dysphonia indicates otherwise. Conversely, it is possible that the psychologic sophistication of some of these patients, who in the present study tended to be well educated and in two instances were engaged in psychotherapy at the time symptoms began, led them toward psychologic explanations. Such explanations may be promoted within a medical milieu in which reductionistic thinking predominates; a little-known disorder without clear physical etiology may be readily dismissed as “in the patient’s head.” In such situations, psychiatrists need to be cautious about undertaking psychotherapeutic treatment of a presumed conversion disorder. There has also been a tendency in the opposite direction to assume that the success of the surgical treatment of the illness implies a purely nonpsy136
chologic etiology. As the success of vagotomy in treating peptic ulcer disease illustrates, this implication is unwarranted. The frequently reported deleterious effect of stress on degree of symptomatology suggests that psychologic factors play some role in the expression of the illness. Moreover, it is possible that the stressful antecedents described by some of the patients in the current study did contribute to the initiation of the illness, but did so via a psychophysiologic rather than a conversion mechanism. What is the psychiatrist’s proper role? Clearly, there is considerable havoc wrought by this illness, and the psychiatrist can help in several ways: most obviously, psychiatrists can treat the secondary depression and anxiety often accompanying the illness, but they can also help by being informed about this disorder so as to prevent psychiatric therapies that may delay more effective surgical treatment. An informed psychiatrist would be able to participate in preoperative evaluation in order to screen for coexisting psychiatric disorders, while being able to appreciate that a physical disorder without a clear organic basis, even in the presence of an apparent psychologic precipitant, cannot be assumed to have a psychologic etiology unless criteria are met for the diagnosis of a somatoform disorder.
References 1.
BrodnitzF: Spastic dysphonia. Ann Otol85:210-214, 1976
2. Dedo H: Recurrent laryngeal nerve section for spastic dysphonia. Ann Otol 85:451-459, 1976
3. Traube, cited by Arnold G: Spastic dysphonia I: 4. 5. 6. 7. 8. 9. 10.
Changing interpretations of a persistent affliction. Logos 2:3-14, 1959 Berendes, cited by Arnold G: Spastic dysphonia I: Changing interpretations of a persistent affliction. Logos 2:3-14, 1959 Izdebski K, Dedo I-k Spastic dysphonia: A patient profile of 200 cases. Am J Otol5:7-14, 1984 Aronson A, Brown J, Litin E, Pearson J: Spastic dysphonia I. Voice, neurologic, and psychiatric aspects. J Speech Hear Dis 33:203-2X3, 1968 Dedo H, Izdebski K: Intermediate results of 306 recurrent laryngeal nerve sections for spastic dysphonia. Laryngoscope 93:9-16,1983 Barton R: Treatment of spastic dysphonia by recurrent laryngeal nerve section. Laryngoscope 89:244 249, 1979 Aronson A, DeSanto L: Adductor spastic dyssphonia: Three years after recurrent laryngeal nerve resection. Laryngoscope 93:1-B, 1983 Biller H, Som M, Lawson W: Laryngeal nerve crush for spastic dysphonia. Ann Otol S&531-532, 1979
Psychiatric Role in Spastic Dysphonia
11. Biller H, Som M, Lawson W: Laryngeal nerve crush for spastic dysphonia. Ann Otol92497, 1983 12. Dedo H, Townsend J, Izdebski K: Current evidence for the organic etiology of spastic dysphonia. Otolaryngology 86:875-880, 1978 13. Feldman M, Nixon J, Finitzo-Hieber T, Freeman F: Abnormal parasympathetic vagal function in patients with spasmodic dysphonia. Ann Intern Med 100:491-495, 1984 14. Schaefer S: Neuropathology of spasmodic dysphonia. Laryngoscope. 93:1183-1201, 1983
15. Diagnostic and Statistical Manual-III. American Psychiatric Press, 1980. 16. Hackett T, Cassem N (eds): Massachusetts General Hospital Handbook of Psychiatry. St. Louis, Mosby, 1978, p 130 Direct reprint requests to: Barry I. Ginsberg, M.D. 10 Muzzey Street Lexington, MA 02173
137
Mt. Sinai Medical Center, New York
Joel J. Wallack, M.D. Division of Consultation-Liaison
Psychiatry,
Beth lsrael Medical Center, New York
James J. Srain, M.D. Division of Liaison Psychiatry,
Mt. Sinai Medical Center, New York
Hugh F. Biller, M.D. Department
of Otolaryngology,
Mt. Sinai Medical Center, New York
Abstract: The authors evaluated 11 surgically-treated patients with spastic dysphonia, a phonation disorder of unclear etiology. The results indicate that the illness does not appear to be a somatoform disorder, but that stress may play a role in its expression, and that there may be secondary depression and anxiety. The experience of spastic dysphonics suggests that psychiatric treatments may be inappropriately applied to an illness without clear organic etiology, whereas, conversely, a proper psychiatric role may be rejected when effective medical or surgical treatment is available. The authors recommend that psychiatrists evaluating patients with illnesses of unclear etiology should be cautious in making a primary psychiatric diagnosis unless DSM-111 criteria are met.
Spastic dysphonia is a phonation disorder of unclear etiology that has not, to our knowledge, been previously reported in the psychiatric literature. Nevertheless, psychotherapy and somatic psychiatric therapies are often among the multitude of treatments these patients receive. The results of psychiatric treatment have been disappointing [l], and the role of psychiatrists in the treatment of these patients has come under attack [2]. This communication reports the results of a study of 11 surgically-treated spastic dysphonics, and seeks to redefine the proper role for psychiatrists in the overall care of patients with this disorder. Spastic dysphonia was first described by Traube in 1871 [3]. Berendes [4] described it as “a disorder of phonation which is characterized by a strained, 132 ISSN 0163~8343/88/$3.50
creaking, and choked vocal attack, a tense and squeezed voice sound that is accompanied by extreme tension of the entire phonatory system,” and likened it to “stuttering with vocal cords.” A recent profile of 200 cases 151, as well as earlier clinical descriptions, indicate that the disorder usually (but not always) begins in middle age, is somewhat more prevalent in women, has a gradual onset, and has a chronic course, remaining without significant change once it has reached a plateau. Although a Medline search was unable to find mention of spastic dysphonia in the psychiatric literature, the otolaryngologic literature has included psychiatric opinions and assumptions. Brodnitz [l] described spastic dysphonia as a conversion disorder “at the opposite end (from hysterical aphonia) of functional voice disorders.” Evidence cited in favor of this etiology includes the recovery of normal phonation while singing and when drugged with sodium amytal or alcohol, as well as the presence in some cases of emotional trauma or stress seemingly related to the onset of symptoms. Congruent with this point of view, psychotherapy and/or speech therapy were thought to be the treatments of choice, although reports of success were lacking. Moreover, Aronson and colleagues [6] had reported in 1968 that MMPI profiles of female spastic dysphonics showed elevation on the depression scale more so than on the hypochondriasis or hysteria scale. This contrasts with General HospitalPsychiatry 10,
132-137,
1988
0 1988 Elsevier Science Publishing Co., Inc. 52 Vanderbilt Ave., New York, NY 10017
Psychiatric Role in Spastic Dysphonia
“conversion V” seen in somatization: the hypochondriasis and hysteria scales are more elevated than is the depression scale. Psychiatric interviews in this study found that spastic dysphonia patients were rigid, conscientious, highly responsible people, but these traits did not differentiate them from other medically ill patients. The interviews generally failed to find distinct precipitating emotional conflict or trauma. Nonetheless, the view of spastic dysphonia as a psychoneurotic symptom predominated until 1976, when a surgical treatment was reported by Dedo [2]. Dedo performed a unilateral section of the recurrent laryngeal nerve (RLN), which paralyzes the vocal cord in the paramedian position, allowing a somewhat breathy, but usually improved, phonation. Subsequently, Dedo [7] reported highly positive results with the procedure on 212 patients affected by spastic dysphonia; other groups [8,9] have reported more mixed but generally positive results. One of the authors (HFB) [lo] described a nerve crush procedure in 1977; however, it was subsequently reported [ll] that the initially favorable results of the procedure did not last, and it was abandoned in favor of the RLN section. Concomitant with these reports, various organic etiologies have been proposed. Dedo [12] has suggested that a virally induced proprioceptive deficit is responsible. Other investigators [13,14] have favored central cranial nerve dysfunction. Since the advent of the surgical appproach, there has been a pendulum swing away from any appreciation of emotional factors in spastic dysphonia, and away from psychiatric involvement in the care of these patients. Dedo [2] reported that the belief in a psychoneurotic etiology “just causes time and money to be wasted on psychotherapy, at least for the vocal disorder itself, and it increases the patient’s guilt and emotional stress.”
Methods We attempted to contact each of the 26 patients who had been treated for spastic dysphonia with unilateral RLN crush and/or section on the otolaryngology service at Mt. Sinai Hospital from 1976 to 1984. Of these patients, 11 agreed to participate; 10 were interviewed in person, and 1, who was located out of state, by telephone. A semistructured interview was used; the patient was encouraged to trace in detail the history of the illness and the treatment received. The interview also included questions about the effect of stress on the vocal
Table 1. Treatment Patient First M.D. Total M.D.s Speech therapy Psychotherapy Hypnosis Biofeedback Acupuncture Psychiatric drugs Chiropractor Meditation/relaxation Psychotherapy Help voice? Psychiatric drugs help voice?
1234567891011 0” 0 PbO F’F F F 4 6 5 4 7 6 5 5 +++++ +++ + +++++ ++ + + + + ++++++ + +
F F F 10 3 10 + + +
NdNNNNN
N
Y’NNNNN
NN
++ +
+
“0, otolaryngologist. ‘I’, psychiatrist. ‘F, family doctor. “N, no.
‘Y, yes.
symptoms, medical and psychiatric history with particular attention to somatoform disorders, and the effect of the illness on work and social life. The subjects included 7 females and 4 males, whose age ranged from 34 to 63 with a mean of 46. The age at the time of onset of symptoms ranged from 29 to 51, with a mean of 36. One patient had completed only high school, three had completed at least 2 years of college, five had been graduated from college, and two had graduate degrees. Eleven were white and one was Hispanic. Eight were married, one divorced, one widowed, and one had never married.
Results The experience of the patients in obtaining treatment (summarized in Table 1) indicates a pattern of widespread difficulty in diagnosis and treatment. Seven patients originally presented to family physicians, three to otolaryngologists, and one to a psychiatrist. Each patient subsequently saw at least three other specialists with a mean of 5.5; patients received a mean of 3.6 different kinds of treatment. Seven patients saw psychiatrists, with one of these patients also seeing a psychologist and a pastoral counselor. They each received psychotherapy as well as psychopharmacotherapy (sometimes from a physician other than the psychiatrist); one other patient who did not see a psychiatrist 133
B. I.
Ginsberg et al.
Table 2. Patients’ evaluation of surgery
Patient RLN crush RLN section
1
2
3
4
5
6
7
8
(-)
(+)
(-)
(+I--)
(-)
(+)
also received psychiatric medication. None of the patients felt that the psychotherapy was helpful for their speech disorder, although the patients frequently commented that their psychotherapy had been helpful in other ways. Seven of the eight patients who received psychiatric medication felt that it had not helped their speech, but one patient reported some improvement with alprazolam. Two of the patients had received a nerve crush procedure, one a crush followed by a section, and the other eight, a nerve section. Subjective evaluations of the effect of surgery upon the voice disorder is summarized in Table 2; all three who had had the crush felt that any benefit was only temporary and, overall, the procedure had not been helpful, the one who subsequently had the section, however, felt positively about that procedure. Of the eight patients who received only the section, four rated it positively, one was equivocal, and three were negative. This is similar to the followup results reported by Aronson and DeSanto [9] in a study of 33 patients, but is less positive than the results reported by Dedo and Izdebski [7], who studied 306 patients, and by Barton [8], who reported on 11 patients. Information germane to psychiatric diagnosis is contained in Table 3. Almost all the subjects reported a major stress (change of job, marital situation, financial situation, illness or death of someone close, major illness, or emotional problems) within the year prior to the onset of vocal symptoms. Most of the subjects reported that once the illness had begun, stress made the symptoms worse. Although four patients had had psychiatric treatment prior to their illness and the same number had psychiatric illness in the family, only one had a previous history of physical symptoms of unclear organic etiology, and none had a history of multiple medical illnesses; indeed, except for a not infrequent history of postnasal drip and other mild respiratory symptoms, the patients described themselves as usually healthy. None of the subjects had personally experienced similar vocal symptoms or had witnessed it in significant figures in their lives; such a “symptom model,” although not a criteron for conversion disorder in DSM-III 1151, 134
9 (-)
10
11
(+)
(+)
has been associated with that diagnosis [16]. Eight subjects reported significant depression (frequently including suicidal thoughts) secondary to the speech disorder, and two reported significant anxiety (usually about being called upon to speak) secondary to the disorder. Although six patients felt that their work status had been adversely affected by the illness and three felt that the illness had adversely affected their general social relations, none of the patients reported on adverse effect on marital relations.
Case Reports Patient 1 was a widow in her early fifties. She had been in psychotherapy since the death of her mother 4 years earlier, when she noted a “strain” and “tension” in her voice that she reported to have occurred after an upsetting session with her psychotherapist. She and her therapist had begun to discuss the possibility of terminating treatment and she was also involved at that time in leaving her home town. Concerns about financial independence were present at that time as well. During the next 6 months, the symptoms gradually worsened; she saw an otolaryngologist who referred her for speech therapy with little result. After the failure of speech therapy, she and her psychiatrist decided to intensively explore the possibility of a psychologic cause, and she resumed psychotherapy, which had been terminated as planned. Regardless of what she felt was progress in understanding, there was no effect on the vocal symptoms. She saw a second otolaryngologist, had another course of speech therapy, saw another psychiatristhypnotherapist (who, she reports, was sure that the vocal problem had a psychologic basis) for hypnosis, all without significant help. At periods of stress she took thioridazine and later trifluoperazine, without effect on the vocal symptoms; she did find that alprazolam provided some improvement for a period of hours. She received a nerve crush 3 years after the onset of symptoms with relief lasting about 6 weeks, then a nerve section 26 months later, resulting in an almost normal voice for the next 2 years until the return of some milder symptoms 6 months prior to the study. There was no history of multiple somatic problems, nor of a symptom model. Patient 2 was in her mid-thirties when symptoms began. She had had a major disappointment in her
Psychiatric
Table 3. Psychiatric
diagnostic
Role in Spastic Dysphonia
information
Patient Major stress <1 yr prior to S.D.” Stress makes voice worse Past somatic ills and/or hypochondria Symptom model Past psychiatric history Family psychiatric history Depression/anxiety 2” to S.D. Work problems 2” to S.D. Marital problems 2” to S.D. Social problems 2” to S.D.
1
2
3
+”
+ +
+
+ + +
+ + +
+
+ +
4
5
6
7
8
9
+
+ +
+ +
+ +
+ +I-’
+ +
+
-t +
-t
+
+
+
+ +
10
+ +
11 + +
+ +
+
+
+
+
+
+
“S.D., spastic dysphonia. ‘C , present. ’ + i - , equivocal.
career and felt dissatisfied with her life, which prompted her to begin psychotherapy 3 months prior to the onset of symptoms. After she was told by an otolaryngologist that she had a normal larynx, she assumed that the voice disorder was an “interesting“ symptom related to the expression of feelings in the new psychotherapy and would disappear spolitaneously. The symptoms did not remit, however, and a stormy psychotherapy was terminated at the suggestion of her psychiatrist after 1 year. She later underwent group psychotherapy, pastoral counseling, two courses of speech therapy, hypnosis, and treatment with diazepam, all without significant effect on her voice disorder. She received a nerve crush some 20 years after the onset of symptoms, with improvement in phonation. However, she disliked the breathy quality to her speech and declined to have nerve section after the symptoms recurred. Patient 3, a successful professional, had the onset of symptoms in his late 20s. Although he could identify no specific environmental or emotional precipitant, he considered himself a “hyper” individual, and the first physician he contacted was a psychiatrist, “to eliminate that possibility.” He reported that the psychiatrist was never sure of the etiology of the speech disorder, but he continued in treatment irregularly during the next 3 years and was treated at various times with diazepam, oxazepam, lithium, and antidepressants, as well as with biofeedback. These treatments were not helpful for the speech disorder. He had three surgical procedures: a recurrent laryngeal nerve crush, with improvement in speech lasting 23 months; a recurrent laryngeal nerve section, with improvement lasting 6-7 months, and a superior laryngeal nerve section, without improvement. Patient 4 was 29 years old when she noticed a “hesitancy” in her voice that progressed into the clinical picture of spastic dysphonia. She reported that the otolaryngologist she saw several months later in-
dicated that she had either multiple sclerosis, myasthenia gravis, or a psychologic problem. She had an inpatient neurologic workup, including a placebo trial (with some relief of symptoms), leading to a referral to a psychiatrist. She began psychiatric treatment that lasted 3 years and uncovered a possible precipitant. About a year and a half before her symptoms began, an event had taken place about which the patient appeared to have had much unconscious guilt. Despite exploration of her guilty feelings, the voice showed no improvement, although she found that her newly gained insights were valuable. At one point, the psychiatrist referred her to another otolaryngologist, who reportedly told her that the problem was psychologic and sent her back to the psychiatrist. She also received speech therapy for 1 year without significant benefit, and was given diazepam, also unsuccessfully. She became depressed, stopped socializing, was hypersomnolent, and became somewhat obese. She received a nerve crush 3 years after the onset of the disorder, with temporary improvement, followed by a nerve section, with more lasting effect, although the voice problem remains at a less severe level.
Discussion Almost invariably, these patients had seen several doctors and received various treatments with poor results prior to surgery. This is consistent with other reports [2,5]; in fact, the first patient to receive a RLN section had seen 29 doctors over 17 years [2]. Most were told that the problem was psychogenic or, on their own, they felt it was psychogenic at some point during the course of the illness. Most received psychiatric treatment, consistently without benefit in terms of the speech
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disorder. Yet, 9 of the 11 patients noted an association of stressful events with the onset of the symptoms, and this may help explain the frequency and persistence of psychiatric treatment in the face of such poor results. As the case reports illustrate, a highly suggestive psychologic precipitant is sometimes presented. Nonetheless, several features of the illness are inconsistent with conversion disorder. The illness usually has a gradual onset and continues uninterrupted for many years; DSM-III notes that conversion disorder “probably is usually of short duration, with abrupt onset and resolution.” In all 11 patients, no symptom model was present. The previously cited study by Aronson and associates [6] found that MMPI and psychiatric interviews did not indicate that these patients tend toward somatization, and the histories of the patients in the present study were inconsistent with a diagnosis of somatization disorder. The interview data, albeit retrospectively, indicate that most patients with this disorder suffer anxiety and depression secondary to the vocal symptoms, and that stress seems to exacerbate the symptoms. The study data suggest that the most appropriate psychiatric diagnosis for most of these patients would be psychologic factors affecting physical condition (DSM-III code 316.00), although during the course of the illness, particularly prior to surgical treatment, some may have warranted a diagnosis of an adjustment disorder or, more rarely, major depression or an anxiety disorder. Although it is possible that further psychiatric exploration with these patients might have led to improvement of the vocal symptoms, the absence of any report of psychotherapeutic success with spastic dysphonia indicates otherwise. Conversely, it is possible that the psychologic sophistication of some of these patients, who in the present study tended to be well educated and in two instances were engaged in psychotherapy at the time symptoms began, led them toward psychologic explanations. Such explanations may be promoted within a medical milieu in which reductionistic thinking predominates; a little-known disorder without clear physical etiology may be readily dismissed as “in the patient’s head.” In such situations, psychiatrists need to be cautious about undertaking psychotherapeutic treatment of a presumed conversion disorder. There has also been a tendency in the opposite direction to assume that the success of the surgical treatment of the illness implies a purely nonpsy136
chologic etiology. As the success of vagotomy in treating peptic ulcer disease illustrates, this implication is unwarranted. The frequently reported deleterious effect of stress on degree of symptomatology suggests that psychologic factors play some role in the expression of the illness. Moreover, it is possible that the stressful antecedents described by some of the patients in the current study did contribute to the initiation of the illness, but did so via a psychophysiologic rather than a conversion mechanism. What is the psychiatrist’s proper role? Clearly, there is considerable havoc wrought by this illness, and the psychiatrist can help in several ways: most obviously, psychiatrists can treat the secondary depression and anxiety often accompanying the illness, but they can also help by being informed about this disorder so as to prevent psychiatric therapies that may delay more effective surgical treatment. An informed psychiatrist would be able to participate in preoperative evaluation in order to screen for coexisting psychiatric disorders, while being able to appreciate that a physical disorder without a clear organic basis, even in the presence of an apparent psychologic precipitant, cannot be assumed to have a psychologic etiology unless criteria are met for the diagnosis of a somatoform disorder.
References 1.
BrodnitzF: Spastic dysphonia. Ann Otol85:210-214, 1976
2. Dedo H: Recurrent laryngeal nerve section for spastic dysphonia. Ann Otol 85:451-459, 1976
3. Traube, cited by Arnold G: Spastic dysphonia I: 4. 5. 6. 7. 8. 9. 10.
Changing interpretations of a persistent affliction. Logos 2:3-14, 1959 Berendes, cited by Arnold G: Spastic dysphonia I: Changing interpretations of a persistent affliction. Logos 2:3-14, 1959 Izdebski K, Dedo I-k Spastic dysphonia: A patient profile of 200 cases. Am J Otol5:7-14, 1984 Aronson A, Brown J, Litin E, Pearson J: Spastic dysphonia I. Voice, neurologic, and psychiatric aspects. J Speech Hear Dis 33:203-2X3, 1968 Dedo H, Izdebski K: Intermediate results of 306 recurrent laryngeal nerve sections for spastic dysphonia. Laryngoscope 93:9-16,1983 Barton R: Treatment of spastic dysphonia by recurrent laryngeal nerve section. Laryngoscope 89:244 249, 1979 Aronson A, DeSanto L: Adductor spastic dyssphonia: Three years after recurrent laryngeal nerve resection. Laryngoscope 93:1-B, 1983 Biller H, Som M, Lawson W: Laryngeal nerve crush for spastic dysphonia. Ann Otol S&531-532, 1979
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11. Biller H, Som M, Lawson W: Laryngeal nerve crush for spastic dysphonia. Ann Otol92497, 1983 12. Dedo H, Townsend J, Izdebski K: Current evidence for the organic etiology of spastic dysphonia. Otolaryngology 86:875-880, 1978 13. Feldman M, Nixon J, Finitzo-Hieber T, Freeman F: Abnormal parasympathetic vagal function in patients with spasmodic dysphonia. Ann Intern Med 100:491-495, 1984 14. Schaefer S: Neuropathology of spasmodic dysphonia. Laryngoscope. 93:1183-1201, 1983
15. Diagnostic and Statistical Manual-III. American Psychiatric Press, 1980. 16. Hackett T, Cassem N (eds): Massachusetts General Hospital Handbook of Psychiatry. St. Louis, Mosby, 1978, p 130 Direct reprint requests to: Barry I. Ginsberg, M.D. 10 Muzzey Street Lexington, MA 02173
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