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Distinguishing psychogenic from neurogenic dysfluency when neurologic and psychologic factors coexist
.I. FLUENCY DISORD. 16 (1991), 3-12
DISTINGUISHING PSYCHOGENIC FROM NEUROGENIC DYSFLUENCY WHEN NEUROLOGIC AND PSYCHOLOGIC FACTORS COEXIST DONNA CLARK TIPPETT Depurtment of Speech-Langunge Pathology, The Good Samaritan Hospital; and Division of Rehabilitation Medicine, School of Medicine, The Johns Hopkins University, Bultimore, Maryland
ARTHUR A. SIEBENS Division of Rehabilitation Baltimore, Maryland
Medicine,
School of Medicine.
The Johns Hopkins University.
Reports of acquired dysfluency emphasize the diagnostic importance of symptom reversibility in distinguishing a psychogenic from a neurogenic etiology. This paper describes the onset of dysfluency in a 23-year-old man who had anoxic encephalopathy with diffuse weakness and spasticity, pseudoseizures, and depression following an episode of status epilepticus. Differential diagnosis was complicated by the presence of both objective neurologic abnormalities and psychologic factors. The history, physical examination and analysis of speech characteristics were insuffIcient to make the diagnosis; however. the effect of a therapeutic trial strongly suggested psychogenicity.
INTRODUCTION may be developmental or acquired. Acquired dysfluency is classified as neurogenic or psychogenic. This paper focuses on the difficulty of distinguishing between neurogenic and psychogenic etiologies if both objective neurologic abnormalities and psychological factors are present. We present a case of acquired dysfluency in a 23-year-old man who had anoxic encephalopathy with diffuse weakness and spasticity, pseudoseizures, and depression after an episode of status epilepticus. The history, physical examination and analysis of speech characteristics were insufficient to make the diagnosis: clarification required observing the effects of treatment. Neurogenic dysfluency is defined as dysfluency due to acquired central
Dysfluency
Address correspondence to Donna Clark Tippett, M.A., Department of Speech-Language The Good Samaritan Hospital, 5601 Loch Raven Boulevard, Baltimore,
Pathology, Marvland
21239.
0 1991 by Elsevier Science Publishing Co., Inc. 655 Avenue of the Americas, New York, NY 10010
3 0094-730x/91/$3.50
4
TIPPETT
and SIEBENS
nervous system (CNS) damage (Canter. 1971). It has been associated with multiple areas of cerebral damage. Although a specific area of damage has not been identified clearly, there is a predominance of references to left anterior cerebral damage (Caplan, 1972; Rosenfield. 1972; Farmer, 197.5; Quinn and Andrews, 1977; Helm, Butler, and Benson, 1978; Rosenbek, Messert, Collins, and Wertz, 1978; Donnan, 1979; Baumgartner & Duffy, 1986; Ludlow, Rosenberg, Salazar, Grafman and Smutok, 1987). Right cerebral hemisphere damage (Fleet and Heilman, 1985: Ardila and Lopez, 1986; Baumgartner and Duffy, 1986; Ludlow et al., 1987) and bilateral hemisphere damage (Helm et al.. 1978; Rosenbek et al., 1978) are reported also. In some cases, the area of cerebral damage is difficult to localize, for example, case #4 in Andrews, Quinn, and Sorby. 1972 and case #7 in Helm et al., 1978. Etiologies in these reports include cerebrovascular disease and head trauma. Rosenbek (1984) and HelmEstabrooks (1986) described neurogenic dysfluency in association with these etiological classes as well as others, such as tumor, progressive neurological diseases, and medication effect. Given the variety of neuropathologies that can cause dysfluency, it is not surprising that characterizing neurogenic dysfluency definitively is difficult. However, many authors identify the following characteristics (Canter, 1971; Helm et al., 1978; Horner and Massey, 1983; Fleet and Heilman, 1985; Ardila and Lopez, 1986; Culatta and Leeper, 1988): 1. 2. 3. 4. 5. 6.
occurrence primarily on initial sounds or syllables; lack of relationship to grammatical function of words; inverse relationship with propositionality: no adaptation effect; absence of secondary features; indifference on the part of the speaker with regard to speech characteristics; and 7. association with other neurologid deficits.
Other characteristics of neurogenic dysfluency are reported less often. These additional characteristics include: increased dysfluency on the sounds “r,” “ h,” and “I” (Canter, 1971); normal melody and appropriate loudness (Horner and Massey, 1983; Fleet and Heilman, 1985); absence of avoidances of communication situations (Ardila and Lopez, 1986); and acute distress resulting in a change regarding beliefs about communication (Culatta and Leeper, 1988). In addition, there are discrepancies regarding characteristics of neurogenic dysfluency. Some characteristics that remain controversial are: greater dysfluency on function than on content words (Caplan, 1972); greater dysfluency under conditions of propositionality (Caplan, 1972); presence of secondary features (Rosenfield, 1972; Baratz and Mesulam, 1981); anxiety about speaking (Rosenfield, 1972); and a positive adaptation effect (Quinn and Andrews, 1977).
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
5
In their retrospective study, Baumgartner and Duffy (1986) described “typical” characteristics of 16 patients with neurogenic dysfluency (mean age = 54 years; 69% over age 50). These patients tended to be righthanded men with average education. Dysfluency began during onset or worsening of neurologic problems, and was often associated with aphasia, apraxia of speech, or dysarthria. When dysfluency was present with other speech-language impairments, onset was simultaneous. The most common dysfluencies were sound/syllable repetitions, word repetitions, and prolongations. Struggle behavior occurred infrequently, although patients were aware of their dysfluencies. Unlike neurogenic dysfluency, psychogenic dysfluency is described infrequently in the literature. Psychogenic dysfluency is defined as nondevelopmental, nonneurogenic dysfluency of sudden onset, temporally related to some form of psychologic stress (Deal, 1982). Rosenbek et al. (1978) reported a possible case of psychogenic dysfluency in a 53-yearold, right-handed man with a right parietal lobe cerebrovascular accident (CVA). A definitive etiology was confounded by the presence of focal neuropathology. Deal (1982) reported a clear case of psychogenic dysfluency in a 28-year-old man who became dysfluent seven days after admission to a psychiatric facility following a suicide attempt. Baumgartner and Duffy (1986) described psychogenic dysfluency in 49 patients (mean age = 46 years; 52% under age 50) who did not have nervous system damage and in 20 patients (mean age = 44 years; 70% under age 50) who did have nervous system damage. In both groups, there was an equal distribution of males and females, typically right-handed with average education. In the first group, neurologic exams were negative, but patients reported CNS-like complaints, for example, headache. Onset of dysfluency was usually simultaneous with the onset or worsening of these CNS-like symptoms. In the second group, etiologies of nervous system disease varied, but often included degenerative processes, closed head injuries, or convulsions. It was not unusual for etiologies to be unknown, or for neurologic examinations to be normal. Localization of neuropathology was also unknown in some instances, but included left- and righthemisphere and posterior fossa infarcts. Deal (1982) and Culatta and Leeper (1988) identified the following characteristics of psychogenic dysfluency: 1. sudden onset; 2. coincidence with an emotional crisis; 3. decrease with general improvement in a patient’s outlook, attitude, and self-concept; 4. minimal or unusual effect of traditional treatment techniques designed for developmental stuttering (e.g., choral reading, white noise, delayed auditory feedback, singing, or exposure to different communicative situations);
TIPPETT
6
5. 6. 7. 8. 9.
atypical dysfluency pattern; high dysfluency rate; no islands of fluency; no secondary features: and disinterest on the part of the patient stuttering.
regarding
and SIEBENS
the concept
of
Baumgartner and Duffy (1986) previously reported that psychogenic dysfluency tends to coincide with the onset of psychiatric difficulties, such as conversion reaction, anxiety neurosis, depression, and personality disorder. In addition, they found that: psychogenic dysfluency usually is not accompanied by other speech-language impairments; sound/syllable repetitions are the most frequent dysfluency type; and symptomatic therapy may result in rapid and dramatic improvement. Unlike Deal (1982) and Culatta and Leeper (1988), Baumgartner and Duffy (1986) observed struggle behavior associated with psychogenic dysfluency. Roth, Aronson, and Davis (1989) expanded the description of psychogenic dysfluency in 12 patients (ages 21-79 years old) originally described by Baumgartner and Duffy (1986). As reported in the earlier study, Roth et al. (1989) stated that: dysfluency was the primary complaint of 11 patients; all 12 patients had additional neurologic-like complaints that were nonorganic; duration of stuttering from onset to examination varied from 4 hr to 1.5 years; the stuttering of most patients was variable in severity and became normal either spontaneously or as a result of speech therapy or psychotherapy. Roth et al. (1989) added that psychologic stress was associated with onset in 10 patients, and that the Minnesota Multiphasic Personality Inventory profiles of 9 out of 10 patients were abnormal, with 4 profiles reflective of a conversion reaction.
PRESENTATION
OF CASE
A 23-year-old right-handed man with a history of refractory generalized tonic-clonic seizures was admitted to an acute care hospital in status epilepticus. The patient was a 32-week premature identical twin with an otherwise unremarkable perinatal history. His twin brother was healthy and seizurefree. The patient’s speech-language development was normal. He first had seizures at age 3 years. Details of early neurologic work-up were not available, but a diagnosis of idiopathic epilepsy was made. During adolescence, the patient received psychotherapy for an unspecified behavioral disorder and depression. Periods of depression coincided with problems related to health, unemployment, and poor social relationships. Over the past 20 years, the patient was treated with various anticonvulsants with variable results, and was hospitalized multiple times for
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
7
uncontrolled seizures. He suffered a depressed skull fracture at age 11 years secondary to a pedestrian-motor vehicle accident. After this accident an increase in his seizure frequency was noted. Eight months prior to this hospital admission, inpatient video/electroencephalography (EEG) monitoring revealed multiple bilateral seizure foci. Seizures consisted of tonic and tonic-clonic activity and drop attacks. Just prior to admission, seizures had been fairly well controlled on valproic acid and carbamazepine. One week prior to admission, the patient’s seizure frequency unexpectedly increased to an average of one generalized seizure each day. Medication noncompliance or intercurrent illness was not suspected. On the day of admission, he had 15 generalized seizures without return of consciousness. In the emergency room, seizures were controlled with 5 mg of diazepam and I gm of phenytoin. Evaluation failed to reveal an etiology for his status epilepticus. Computed tomography (CT) scan was normal. Over the next several days, although he had only sporadic seizures, his mental status failed to improve. Neurologic examination revealed a minimal, but symmetrical, withdrawal response and grimace to painful stimuli. Cranial nerve exam was within normal limits. Deep tendon reflexes were normal with downgoing toes. However, bilateral ankle clonus was noted. The only abnormal reflex elicited was a sucking response to peri-oral stimulation. EEG during this period revealed a loss of normal background rhythm, irregular diffuse slow activity, and bilateral temporal sharp waves. This was consistent with a diffuse encephalopathy and potential bilateral temporal seizure foci. Despite improved control of his seizures, his post-ictal level of consciousness necessitated intubation and mechanical ventilation for airway protection. His intensive care unit (ICU) course was complicated by intravenous line sepsis and a urinary tract infection. His anticonvulsants were changed to phenytoin and carbamazepine. After 17 days in the ICU, seizures were controlled and mental status improved. The patient was transferred to the neurology ward. Neurologic examination was nonfocal, except for confusion, severe stuttering, and spasticity. These tindings were thought to be secondary to anoxic encephalopathy. While on the ward, he had repeated episodes of head-bobbing, nodding, and facial contortions that differed from earlier seizures. Repeat EEG monitoring during these episodes failed to reveal any seizure activity. This was consistent with a diagnosis of psychogenic seizures. Five weeks postonset, he was transferred to our inpatient rehabilitation unit. At admission to the rehabilitation unit, the patient’s neurologic examination revealed severe stuttering, mild bilateral lower extremity increased tone, mild cerebellar dysmetria, and ataxia. Barthel Index (Mahoney and Barthel, 1965) was 53/100. The patient was referred for occupational therapy, physical therapy, and speech-language pathology.
TIPPETT and SIEBENS
8 A psychiatrist psychogenic.
was consulted
SPEECH-LANGUAGE
who felt that the patient’s
PATHOLOGY
dysfluency
was
EVALUATION
A variety of tests were administered including the Wechsler Memory Scale (Wechsler and Stone, 1943, Mayo Orientation and General Information Assessment (Wertz, 1985), Complex Ideational Material of the Boston Diagnostic Aphasia Examination (Goodglass and Kaplan, 1976) and Modified Token Test (DeRenzi and Faglioni, 1978). Test results revealed normal cognitive and receptive language abilities. The patient was alert and oriented. He followed commands containing up to six informational units and complex syntax. He correctly answered questions about orally presented paragraph length information. Verbal expressive language was characterized by intact vocabulary and syntax with dialectical variations. Oral peripheral examination was unremarkable. There was severe dysfluency with sound/syllable, part-word, or whole-word repetitions occurring on almost every word. The patient’s speech had characteristics of neurogenic dysfluency. Specifically, speech was characterized by repetitions, not only of initial sounds but of final sounds as well. Also, there were whole-word repetitions of single syllable words and part-word repetitions of multisyllable words. Dysfluencies occurred on small grammatical words as well as substantive words. Dysfluency persisted across automatic speech, reading, recitation, and monolog tasks. An adaptation effect was not observed. There were no secondary features, and the patient stated that he was not anxious. Characteristics of psychogenic dysfluency were also present. The onset of dysfluency was related to an identifiable emotional crisis and possibly cumulative psychologic stress. Response to treatment techniques was unusual. Although fluency improved during rhythmic choral reading, the patient’s phonatory quality became tremulous and his head shook slightly from side-to-side, similar to head titubation. These behaviors were not observed prior to the choral reading exercise or afterwards. The patient was fluent when whispering test stimuli to himself, despite an otherwise high dysfluency rate and absence of islands of fluency. He did not demonstrate interest in the concept of stuttering. Thus, analysis of speech characteristics was insufficient to determine if dysfluency was neurogenic or psychogenic, inasmuch as characteristics of both were present. Psychogenicity was considered tentatively at this point because: the onset of dysfluency coincided with a stressful event (i.e., hospitalization for status epilepticus); psychogenic seizures occurred; other speech-language impairments were absent; and there was a history of psychological difficulties. An organic basis for the patient’s
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
9
dysfluency could not be ruled out, however, in light of the seizure history, onset following status epilepticus, and abnormal neurologic examination. HOSPITAL COURSE AND SPEECH-LANGUAGE PATHOLOGY TREATMENT Speech-language pathology treatment was initiated using rhythmic speaking. Remarkably, fluency improved within the first session. The patient was instructed to practice the technique of clapping or tapping in time with speaking over the weekend. During that weekend, the patient had repeated episodes of head thrashing, leg kicking, arm waving, and facial twitching for periods of up to 2 hr. These episodes were not similar to his previous epileptic seizures and were clinically consistent with psychogenic seizures. Following these episodes, the patient was alert, but mute and did not ambulate. Neurologic examination was otherwise normal and comprehension was felt to be intact. On the subsequent Monday, a conference was held with the patient, attending physician, resident, and speech-language pathologist to discuss the events of the weekend. It was emphasized that there were no medical or physiologic abnormalities which could account for the patient’s motor impairments in speaking and walking. He was reassured that no lifethreatening health problems were at issue. He was gently but firmly confronted with the possibility that his will, in the context of the supportive environment of the rehabilitation setting, could overcome his problems with communication and ambulation. During the conference, fluency improved relative to that demonstrated over the preceding weekend. Following the conference, use of rhythmic speaking was reinitiated during a speech-language pathology treatment session. Gradually, clapping and tapping were extinguished over a period of days. Caregivers facilitated carryover of fluency by verbal support and cues to “use speech like that in therapy.” Fluency, mobility, and affect improved concurrently. Two weeks after admission, the patient was fluent, ambulatory, and demonstrated a positive outlook. He had no further seizure episodes or neurologic symptoms. Bat-the1 Index (Mahoney and Barthel, 1965) was 98/100 at discharge. The patient was discharged home on phenytoin and carbamazepine. He was referred to a community mental health facility for follow-up, to a boarding house for alternate living arrangements, and to vocational rehabilitation for employment. Dysfluency had not returned three months after discharge. DISCUSSION A therapeutic trial strongly suggested that dysfluency was psychogenic. The diagnosis was clear once rapid resolution occurred. Rapid resolution of dysfluency was seen also in patients described by Rosenbek and his
IO
TIPPETT
and SIEBENS
colleagues (1978), Deal (1982), Baumgartner and Duffy ( 1986) and Roth and her colleagues (1989). Rosenbek et al.‘s (1978) patient used syllabletimed speech to decrease rate and became fluent in six sessions (96 hr). Deal’s (1982) patient became fluent in the first treatment session while using delayed auditory feedback; he achieved carryover in 3 weeks. Baumgartner and Duffy (1986) found that psychogenic dysfluency completely or nearly disappeared following one to two symptomatic speech therapy sessions even though there was no improvement in patients’ overall neurologic status. In Roth et al.‘s (1989) report, the speech of 1I of their 12 patients became normal or nearly normal during a “brief time” (measured in minutes to days where indicated) at their clinic. Five patients became fluent with speech therapy; four became fluent spontaneously; one became fluent with group psychotherapy, and another while discussing the onset of dysfluency. In contrast, neurogenic dysfluency has been reported to persist for several weeks to several years (Andrews, Quinn, and Sorby, 1972; Quinn and Andrews, 1977; Helm et al., 1978; Rosenbek et al., 1978; Donnan, 1979; Fleet and Heilman, 1985; Ardila and Lopez, 1986; Ludlow et al., 1987). Provision of and response to treatment are not specified in all reports of neurogenic dysfluency. However, Rosenbek et al. (1978) reported that two patients did not demonstrate a change in dysfluency 1I weeks after evaluation following speech-language pathology treatment: two others did not experience a change in dysfluency without treatment (one at six months follow-up; one at one year follow-up); one patient showed some improvement in his dysarthria and dysfluency after two weeks of symptomatic treatment for dysarthria. We conclude that quick responsiveness to treatment is an important criterion in the differential diagnosis of acquired dysfluency. This conclusion is consistent with previous reports. Baumgartner and Duffy (1986) recognized that rapid, dramatic improvement in fluency following symptomatic therapy helps to confirm a psychogenic etiology. Roth et al. (1989) wrote that “the most revealing diagnostic indicator . . is the patient’s improvement with speech therapy, during psychosocial interview, or with psychotherapy; such improvement is a diagnostic indicator of psychogenicity” (p. 642). Sapir and Aronson (1990) stated that “the strongest evidence for psychogenic etiology is symptom reversibility (i.e., the immediate removal of symptoms by therapy, and/or sudden onset. disappearance, and recurrence of symptoms without concurrent changes in the patient’s medical status)” (p. 506). Our treatment consisted of: providing daily speech-language pathology treatment; identifying a context compatible with fluency; providing a supportive, caring environment; and utilizing frank confrontation. an approach that is not traditionally used with dysfluent patients. Our intervention also included therapy for mobility impairments and provision of
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
11
follow-up services regarding mental health, employment, and living circumstances. Aspects of our approach have been identified as appropriate by previous investigators. Roth et al. (1989) advocated that patients with psychogenic dysfluency be shown how to increase fluency through traditional techniques and be persuaded that they have greater than expected capacity for fluent speech. They also stated that encouragement and optimism on the part of the clinician are essential, and that psychotherapy may be indicated if conflicts persist. Sapir and Aronson (1990) advised
intensive speech therapy and in-depth interview, allowing patients to ventilate feelings, in order to reduce speech symptoms. These observations underscore the reversibility of psychogenic dysfluency and the success of speech-language pathology treatment combined with counseling and supportive therapy. The authors gratefully acknowledge MS Patricia Linden Castelli, M.A., Director, Department of Speech-Language Pathology, The Good Samaritan Hospital, for her review of this manuscript, as well as all of the members of the Department of Speech-Language Pathology for their comments on an earlier version of this paper. The authors also wish to thank MS Alexandra Brzozowski for her excellent manuscript preparation.
REFERENCES Andrews, G., Quinn, P.T., and Sorby, W.A. (1982) Stuttering: An investigation into cerebral dominance for speech. Journal of Neurology, Neurosurgery and Psychiatry, 35, 414-418.
Ardila, A., and Lopez, M.V. (1986) Severe stuttering associated with right hemisphere lesion. Brain and Language, 27, 239-246. Baratz, R., and Mesulam, M. (1981) Adult-onset vulsants. Archives of Neurology, 38, 132.
stuttering treated with anticon-
Baumgartner, J., and Duffy, J.R. (1986) Adult onset stuttering: Psychogenic and/ or neurogenic. Miniseminar presented at the American Speech-Language-Hearing Association, Detroit, MI. Canter, G.J. (1971) Observations on neurogenic stuttering: A contribution to differential diagnosis. British Journal of Communication Disorders, 6, 139-143. Caplan, L. (1972) An investigation of some aspects of stuttering-like speech in adult dysphasic subjects. Journal of the South African Speech and Hearing Association, 19, 52-66. Culatta, R., and Leeper, L. (1988) Dysfluency Speech
and Hearing Disorders,
Deal, J.L. (1982) Sudden onset of stuttering: and Hearing Disorders,
isn’t always stuttering. Journal of
53, 486-487.
A case report. Journal
of Speech
47, 301-304.
DeRenzi, E., and Faglioni, P. (1978) Normative data and screening power of a shortened version of the Token Test. Cortex, 14, 41-49.
12
TIPPETT
Donnan, G.A. (1979) Stuttering ofAustralia. 1, 44-45.
as a manifestation
Farmer. A. (1975) Stuttering repetitions adults. Cortex, 11, 391-396. Fleet, W.S.. and Heilman. K.M. sphere lesion in a right-hander.
of stroke.
in aphasic
The Medical Journal
and nonaphasic
brain damaged
(1985) Acquired stuttering from a right hemiNeurology, 35, 1343-1346.
Goodglass, H., and Kaplan, E. (1976) The usxssment disorders. Philadelphia: Lea and Febiger. Helm, N.A., Butler, R.B., rology, 28, 1159-l 165.
and SIEBENS
and Benson,
D.F.
of aphnsicr and related
(1978) Acquired
stuttering.
Near-
Helm-Estabrooks, N. (1986) Diagnosis and management of neurogenic stuttering in adults. In: The Atypical Stutterer: Principles und Practices ofRehabilitation (St. Louis, K. ed.). Orlando, FL: Academic Press. Horner, J., and Massey, E.W. (1983) Progressive dysfluency hemisphere disease. Brain and Langurge, 18, 71-85.
associated
with right
Ludlow, C.L., Rosenberg, J., Salazar, A., Grafman. J.. and Smutok. Site of penetrating brain lesions causing chronic acquired stuttering. Neurology. 22. 60-66.
M. (1987) Annals of
Mahoney, F.I.. and Barthel. D.W. (1965) Functional index. Muryland State Medical Journal, 14. 61-65.
evaluation:
The Barthel
Quinn, P.T., and Andrews, G. (1977) Neurological stuttering-A clinical Journal ofNeurology, Neurosurgery and Psychicrtry, 40. 699-701
entity’!
Rosenbek. J.C. (1984) Stuttering secondary to nervous system damage. In: Nuture and Treatment ofStuttering: New Directions (Curlee, R.F. and Perkins. W.H. eds.). San Diego: College Hill. Rosenbek, J., Messert, lowing brain damage.
B., Collins, M., and Wertz, R.T. (1978) Stuttering Bruin and Language, 6, 82-96.
Rosenfield, D.B. (1972) Stuttering ofMedicine, 287, 991.
and cerebral
ischemia.
New England
fol-
Journal
Roth, C.R., Aronson, A.E., and Davis, L.J. (1989) Clinical studies in psychogenic stuttering of adult onset. Journal of Speech lrnd Hearing Disorders, 54, 634-646. Sapir, S., and Aronson, A.E. (1990) The relationship between psychopathology and speech and language disorders in neurologic patients. Journal of Speech and Hearing Disorders, 55, 503-509. Wechsler, D., Psychological
and Stone, Corporation.
C.
(1945)
Wechsler
memory
scale.
New
York:
Wertz, R.T. (1985) Neuropathologies of speech and language: An introduction to patient management. In: Clinical Management of Neurogenic Communicative Disorders (Johns, D.F. ed.). Boston: Little, Brown and Company. Manuscript received September revised April 1991; accepted May 1991.
1990,
DISTINGUISHING PSYCHOGENIC FROM NEUROGENIC DYSFLUENCY WHEN NEUROLOGIC AND PSYCHOLOGIC FACTORS COEXIST DONNA CLARK TIPPETT Depurtment of Speech-Langunge Pathology, The Good Samaritan Hospital; and Division of Rehabilitation Medicine, School of Medicine, The Johns Hopkins University, Bultimore, Maryland
ARTHUR A. SIEBENS Division of Rehabilitation Baltimore, Maryland
Medicine,
School of Medicine.
The Johns Hopkins University.
Reports of acquired dysfluency emphasize the diagnostic importance of symptom reversibility in distinguishing a psychogenic from a neurogenic etiology. This paper describes the onset of dysfluency in a 23-year-old man who had anoxic encephalopathy with diffuse weakness and spasticity, pseudoseizures, and depression following an episode of status epilepticus. Differential diagnosis was complicated by the presence of both objective neurologic abnormalities and psychologic factors. The history, physical examination and analysis of speech characteristics were insuffIcient to make the diagnosis; however. the effect of a therapeutic trial strongly suggested psychogenicity.
INTRODUCTION may be developmental or acquired. Acquired dysfluency is classified as neurogenic or psychogenic. This paper focuses on the difficulty of distinguishing between neurogenic and psychogenic etiologies if both objective neurologic abnormalities and psychological factors are present. We present a case of acquired dysfluency in a 23-year-old man who had anoxic encephalopathy with diffuse weakness and spasticity, pseudoseizures, and depression after an episode of status epilepticus. The history, physical examination and analysis of speech characteristics were insufficient to make the diagnosis: clarification required observing the effects of treatment. Neurogenic dysfluency is defined as dysfluency due to acquired central
Dysfluency
Address correspondence to Donna Clark Tippett, M.A., Department of Speech-Language The Good Samaritan Hospital, 5601 Loch Raven Boulevard, Baltimore,
Pathology, Marvland
21239.
0 1991 by Elsevier Science Publishing Co., Inc. 655 Avenue of the Americas, New York, NY 10010
3 0094-730x/91/$3.50
4
TIPPETT
and SIEBENS
nervous system (CNS) damage (Canter. 1971). It has been associated with multiple areas of cerebral damage. Although a specific area of damage has not been identified clearly, there is a predominance of references to left anterior cerebral damage (Caplan, 1972; Rosenfield. 1972; Farmer, 197.5; Quinn and Andrews, 1977; Helm, Butler, and Benson, 1978; Rosenbek, Messert, Collins, and Wertz, 1978; Donnan, 1979; Baumgartner & Duffy, 1986; Ludlow, Rosenberg, Salazar, Grafman and Smutok, 1987). Right cerebral hemisphere damage (Fleet and Heilman, 1985: Ardila and Lopez, 1986; Baumgartner and Duffy, 1986; Ludlow et al., 1987) and bilateral hemisphere damage (Helm et al.. 1978; Rosenbek et al., 1978) are reported also. In some cases, the area of cerebral damage is difficult to localize, for example, case #4 in Andrews, Quinn, and Sorby. 1972 and case #7 in Helm et al., 1978. Etiologies in these reports include cerebrovascular disease and head trauma. Rosenbek (1984) and HelmEstabrooks (1986) described neurogenic dysfluency in association with these etiological classes as well as others, such as tumor, progressive neurological diseases, and medication effect. Given the variety of neuropathologies that can cause dysfluency, it is not surprising that characterizing neurogenic dysfluency definitively is difficult. However, many authors identify the following characteristics (Canter, 1971; Helm et al., 1978; Horner and Massey, 1983; Fleet and Heilman, 1985; Ardila and Lopez, 1986; Culatta and Leeper, 1988): 1. 2. 3. 4. 5. 6.
occurrence primarily on initial sounds or syllables; lack of relationship to grammatical function of words; inverse relationship with propositionality: no adaptation effect; absence of secondary features; indifference on the part of the speaker with regard to speech characteristics; and 7. association with other neurologid deficits.
Other characteristics of neurogenic dysfluency are reported less often. These additional characteristics include: increased dysfluency on the sounds “r,” “ h,” and “I” (Canter, 1971); normal melody and appropriate loudness (Horner and Massey, 1983; Fleet and Heilman, 1985); absence of avoidances of communication situations (Ardila and Lopez, 1986); and acute distress resulting in a change regarding beliefs about communication (Culatta and Leeper, 1988). In addition, there are discrepancies regarding characteristics of neurogenic dysfluency. Some characteristics that remain controversial are: greater dysfluency on function than on content words (Caplan, 1972); greater dysfluency under conditions of propositionality (Caplan, 1972); presence of secondary features (Rosenfield, 1972; Baratz and Mesulam, 1981); anxiety about speaking (Rosenfield, 1972); and a positive adaptation effect (Quinn and Andrews, 1977).
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
5
In their retrospective study, Baumgartner and Duffy (1986) described “typical” characteristics of 16 patients with neurogenic dysfluency (mean age = 54 years; 69% over age 50). These patients tended to be righthanded men with average education. Dysfluency began during onset or worsening of neurologic problems, and was often associated with aphasia, apraxia of speech, or dysarthria. When dysfluency was present with other speech-language impairments, onset was simultaneous. The most common dysfluencies were sound/syllable repetitions, word repetitions, and prolongations. Struggle behavior occurred infrequently, although patients were aware of their dysfluencies. Unlike neurogenic dysfluency, psychogenic dysfluency is described infrequently in the literature. Psychogenic dysfluency is defined as nondevelopmental, nonneurogenic dysfluency of sudden onset, temporally related to some form of psychologic stress (Deal, 1982). Rosenbek et al. (1978) reported a possible case of psychogenic dysfluency in a 53-yearold, right-handed man with a right parietal lobe cerebrovascular accident (CVA). A definitive etiology was confounded by the presence of focal neuropathology. Deal (1982) reported a clear case of psychogenic dysfluency in a 28-year-old man who became dysfluent seven days after admission to a psychiatric facility following a suicide attempt. Baumgartner and Duffy (1986) described psychogenic dysfluency in 49 patients (mean age = 46 years; 52% under age 50) who did not have nervous system damage and in 20 patients (mean age = 44 years; 70% under age 50) who did have nervous system damage. In both groups, there was an equal distribution of males and females, typically right-handed with average education. In the first group, neurologic exams were negative, but patients reported CNS-like complaints, for example, headache. Onset of dysfluency was usually simultaneous with the onset or worsening of these CNS-like symptoms. In the second group, etiologies of nervous system disease varied, but often included degenerative processes, closed head injuries, or convulsions. It was not unusual for etiologies to be unknown, or for neurologic examinations to be normal. Localization of neuropathology was also unknown in some instances, but included left- and righthemisphere and posterior fossa infarcts. Deal (1982) and Culatta and Leeper (1988) identified the following characteristics of psychogenic dysfluency: 1. sudden onset; 2. coincidence with an emotional crisis; 3. decrease with general improvement in a patient’s outlook, attitude, and self-concept; 4. minimal or unusual effect of traditional treatment techniques designed for developmental stuttering (e.g., choral reading, white noise, delayed auditory feedback, singing, or exposure to different communicative situations);
TIPPETT
6
5. 6. 7. 8. 9.
atypical dysfluency pattern; high dysfluency rate; no islands of fluency; no secondary features: and disinterest on the part of the patient stuttering.
regarding
and SIEBENS
the concept
of
Baumgartner and Duffy (1986) previously reported that psychogenic dysfluency tends to coincide with the onset of psychiatric difficulties, such as conversion reaction, anxiety neurosis, depression, and personality disorder. In addition, they found that: psychogenic dysfluency usually is not accompanied by other speech-language impairments; sound/syllable repetitions are the most frequent dysfluency type; and symptomatic therapy may result in rapid and dramatic improvement. Unlike Deal (1982) and Culatta and Leeper (1988), Baumgartner and Duffy (1986) observed struggle behavior associated with psychogenic dysfluency. Roth, Aronson, and Davis (1989) expanded the description of psychogenic dysfluency in 12 patients (ages 21-79 years old) originally described by Baumgartner and Duffy (1986). As reported in the earlier study, Roth et al. (1989) stated that: dysfluency was the primary complaint of 11 patients; all 12 patients had additional neurologic-like complaints that were nonorganic; duration of stuttering from onset to examination varied from 4 hr to 1.5 years; the stuttering of most patients was variable in severity and became normal either spontaneously or as a result of speech therapy or psychotherapy. Roth et al. (1989) added that psychologic stress was associated with onset in 10 patients, and that the Minnesota Multiphasic Personality Inventory profiles of 9 out of 10 patients were abnormal, with 4 profiles reflective of a conversion reaction.
PRESENTATION
OF CASE
A 23-year-old right-handed man with a history of refractory generalized tonic-clonic seizures was admitted to an acute care hospital in status epilepticus. The patient was a 32-week premature identical twin with an otherwise unremarkable perinatal history. His twin brother was healthy and seizurefree. The patient’s speech-language development was normal. He first had seizures at age 3 years. Details of early neurologic work-up were not available, but a diagnosis of idiopathic epilepsy was made. During adolescence, the patient received psychotherapy for an unspecified behavioral disorder and depression. Periods of depression coincided with problems related to health, unemployment, and poor social relationships. Over the past 20 years, the patient was treated with various anticonvulsants with variable results, and was hospitalized multiple times for
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
7
uncontrolled seizures. He suffered a depressed skull fracture at age 11 years secondary to a pedestrian-motor vehicle accident. After this accident an increase in his seizure frequency was noted. Eight months prior to this hospital admission, inpatient video/electroencephalography (EEG) monitoring revealed multiple bilateral seizure foci. Seizures consisted of tonic and tonic-clonic activity and drop attacks. Just prior to admission, seizures had been fairly well controlled on valproic acid and carbamazepine. One week prior to admission, the patient’s seizure frequency unexpectedly increased to an average of one generalized seizure each day. Medication noncompliance or intercurrent illness was not suspected. On the day of admission, he had 15 generalized seizures without return of consciousness. In the emergency room, seizures were controlled with 5 mg of diazepam and I gm of phenytoin. Evaluation failed to reveal an etiology for his status epilepticus. Computed tomography (CT) scan was normal. Over the next several days, although he had only sporadic seizures, his mental status failed to improve. Neurologic examination revealed a minimal, but symmetrical, withdrawal response and grimace to painful stimuli. Cranial nerve exam was within normal limits. Deep tendon reflexes were normal with downgoing toes. However, bilateral ankle clonus was noted. The only abnormal reflex elicited was a sucking response to peri-oral stimulation. EEG during this period revealed a loss of normal background rhythm, irregular diffuse slow activity, and bilateral temporal sharp waves. This was consistent with a diffuse encephalopathy and potential bilateral temporal seizure foci. Despite improved control of his seizures, his post-ictal level of consciousness necessitated intubation and mechanical ventilation for airway protection. His intensive care unit (ICU) course was complicated by intravenous line sepsis and a urinary tract infection. His anticonvulsants were changed to phenytoin and carbamazepine. After 17 days in the ICU, seizures were controlled and mental status improved. The patient was transferred to the neurology ward. Neurologic examination was nonfocal, except for confusion, severe stuttering, and spasticity. These tindings were thought to be secondary to anoxic encephalopathy. While on the ward, he had repeated episodes of head-bobbing, nodding, and facial contortions that differed from earlier seizures. Repeat EEG monitoring during these episodes failed to reveal any seizure activity. This was consistent with a diagnosis of psychogenic seizures. Five weeks postonset, he was transferred to our inpatient rehabilitation unit. At admission to the rehabilitation unit, the patient’s neurologic examination revealed severe stuttering, mild bilateral lower extremity increased tone, mild cerebellar dysmetria, and ataxia. Barthel Index (Mahoney and Barthel, 1965) was 53/100. The patient was referred for occupational therapy, physical therapy, and speech-language pathology.
TIPPETT and SIEBENS
8 A psychiatrist psychogenic.
was consulted
SPEECH-LANGUAGE
who felt that the patient’s
PATHOLOGY
dysfluency
was
EVALUATION
A variety of tests were administered including the Wechsler Memory Scale (Wechsler and Stone, 1943, Mayo Orientation and General Information Assessment (Wertz, 1985), Complex Ideational Material of the Boston Diagnostic Aphasia Examination (Goodglass and Kaplan, 1976) and Modified Token Test (DeRenzi and Faglioni, 1978). Test results revealed normal cognitive and receptive language abilities. The patient was alert and oriented. He followed commands containing up to six informational units and complex syntax. He correctly answered questions about orally presented paragraph length information. Verbal expressive language was characterized by intact vocabulary and syntax with dialectical variations. Oral peripheral examination was unremarkable. There was severe dysfluency with sound/syllable, part-word, or whole-word repetitions occurring on almost every word. The patient’s speech had characteristics of neurogenic dysfluency. Specifically, speech was characterized by repetitions, not only of initial sounds but of final sounds as well. Also, there were whole-word repetitions of single syllable words and part-word repetitions of multisyllable words. Dysfluencies occurred on small grammatical words as well as substantive words. Dysfluency persisted across automatic speech, reading, recitation, and monolog tasks. An adaptation effect was not observed. There were no secondary features, and the patient stated that he was not anxious. Characteristics of psychogenic dysfluency were also present. The onset of dysfluency was related to an identifiable emotional crisis and possibly cumulative psychologic stress. Response to treatment techniques was unusual. Although fluency improved during rhythmic choral reading, the patient’s phonatory quality became tremulous and his head shook slightly from side-to-side, similar to head titubation. These behaviors were not observed prior to the choral reading exercise or afterwards. The patient was fluent when whispering test stimuli to himself, despite an otherwise high dysfluency rate and absence of islands of fluency. He did not demonstrate interest in the concept of stuttering. Thus, analysis of speech characteristics was insufficient to determine if dysfluency was neurogenic or psychogenic, inasmuch as characteristics of both were present. Psychogenicity was considered tentatively at this point because: the onset of dysfluency coincided with a stressful event (i.e., hospitalization for status epilepticus); psychogenic seizures occurred; other speech-language impairments were absent; and there was a history of psychological difficulties. An organic basis for the patient’s
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
9
dysfluency could not be ruled out, however, in light of the seizure history, onset following status epilepticus, and abnormal neurologic examination. HOSPITAL COURSE AND SPEECH-LANGUAGE PATHOLOGY TREATMENT Speech-language pathology treatment was initiated using rhythmic speaking. Remarkably, fluency improved within the first session. The patient was instructed to practice the technique of clapping or tapping in time with speaking over the weekend. During that weekend, the patient had repeated episodes of head thrashing, leg kicking, arm waving, and facial twitching for periods of up to 2 hr. These episodes were not similar to his previous epileptic seizures and were clinically consistent with psychogenic seizures. Following these episodes, the patient was alert, but mute and did not ambulate. Neurologic examination was otherwise normal and comprehension was felt to be intact. On the subsequent Monday, a conference was held with the patient, attending physician, resident, and speech-language pathologist to discuss the events of the weekend. It was emphasized that there were no medical or physiologic abnormalities which could account for the patient’s motor impairments in speaking and walking. He was reassured that no lifethreatening health problems were at issue. He was gently but firmly confronted with the possibility that his will, in the context of the supportive environment of the rehabilitation setting, could overcome his problems with communication and ambulation. During the conference, fluency improved relative to that demonstrated over the preceding weekend. Following the conference, use of rhythmic speaking was reinitiated during a speech-language pathology treatment session. Gradually, clapping and tapping were extinguished over a period of days. Caregivers facilitated carryover of fluency by verbal support and cues to “use speech like that in therapy.” Fluency, mobility, and affect improved concurrently. Two weeks after admission, the patient was fluent, ambulatory, and demonstrated a positive outlook. He had no further seizure episodes or neurologic symptoms. Bat-the1 Index (Mahoney and Barthel, 1965) was 98/100 at discharge. The patient was discharged home on phenytoin and carbamazepine. He was referred to a community mental health facility for follow-up, to a boarding house for alternate living arrangements, and to vocational rehabilitation for employment. Dysfluency had not returned three months after discharge. DISCUSSION A therapeutic trial strongly suggested that dysfluency was psychogenic. The diagnosis was clear once rapid resolution occurred. Rapid resolution of dysfluency was seen also in patients described by Rosenbek and his
IO
TIPPETT
and SIEBENS
colleagues (1978), Deal (1982), Baumgartner and Duffy ( 1986) and Roth and her colleagues (1989). Rosenbek et al.‘s (1978) patient used syllabletimed speech to decrease rate and became fluent in six sessions (96 hr). Deal’s (1982) patient became fluent in the first treatment session while using delayed auditory feedback; he achieved carryover in 3 weeks. Baumgartner and Duffy (1986) found that psychogenic dysfluency completely or nearly disappeared following one to two symptomatic speech therapy sessions even though there was no improvement in patients’ overall neurologic status. In Roth et al.‘s (1989) report, the speech of 1I of their 12 patients became normal or nearly normal during a “brief time” (measured in minutes to days where indicated) at their clinic. Five patients became fluent with speech therapy; four became fluent spontaneously; one became fluent with group psychotherapy, and another while discussing the onset of dysfluency. In contrast, neurogenic dysfluency has been reported to persist for several weeks to several years (Andrews, Quinn, and Sorby, 1972; Quinn and Andrews, 1977; Helm et al., 1978; Rosenbek et al., 1978; Donnan, 1979; Fleet and Heilman, 1985; Ardila and Lopez, 1986; Ludlow et al., 1987). Provision of and response to treatment are not specified in all reports of neurogenic dysfluency. However, Rosenbek et al. (1978) reported that two patients did not demonstrate a change in dysfluency 1I weeks after evaluation following speech-language pathology treatment: two others did not experience a change in dysfluency without treatment (one at six months follow-up; one at one year follow-up); one patient showed some improvement in his dysarthria and dysfluency after two weeks of symptomatic treatment for dysarthria. We conclude that quick responsiveness to treatment is an important criterion in the differential diagnosis of acquired dysfluency. This conclusion is consistent with previous reports. Baumgartner and Duffy (1986) recognized that rapid, dramatic improvement in fluency following symptomatic therapy helps to confirm a psychogenic etiology. Roth et al. (1989) wrote that “the most revealing diagnostic indicator . . is the patient’s improvement with speech therapy, during psychosocial interview, or with psychotherapy; such improvement is a diagnostic indicator of psychogenicity” (p. 642). Sapir and Aronson (1990) stated that “the strongest evidence for psychogenic etiology is symptom reversibility (i.e., the immediate removal of symptoms by therapy, and/or sudden onset. disappearance, and recurrence of symptoms without concurrent changes in the patient’s medical status)” (p. 506). Our treatment consisted of: providing daily speech-language pathology treatment; identifying a context compatible with fluency; providing a supportive, caring environment; and utilizing frank confrontation. an approach that is not traditionally used with dysfluent patients. Our intervention also included therapy for mobility impairments and provision of
PSYCHOGENIC
FROM NEUROGENIC
DYSFLUENCY
11
follow-up services regarding mental health, employment, and living circumstances. Aspects of our approach have been identified as appropriate by previous investigators. Roth et al. (1989) advocated that patients with psychogenic dysfluency be shown how to increase fluency through traditional techniques and be persuaded that they have greater than expected capacity for fluent speech. They also stated that encouragement and optimism on the part of the clinician are essential, and that psychotherapy may be indicated if conflicts persist. Sapir and Aronson (1990) advised
intensive speech therapy and in-depth interview, allowing patients to ventilate feelings, in order to reduce speech symptoms. These observations underscore the reversibility of psychogenic dysfluency and the success of speech-language pathology treatment combined with counseling and supportive therapy. The authors gratefully acknowledge MS Patricia Linden Castelli, M.A., Director, Department of Speech-Language Pathology, The Good Samaritan Hospital, for her review of this manuscript, as well as all of the members of the Department of Speech-Language Pathology for their comments on an earlier version of this paper. The authors also wish to thank MS Alexandra Brzozowski for her excellent manuscript preparation.
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