Acquired stuttering and bilateral cerebral disease

J. FLUENCY DISORD. 12 (1987), 141-146

ACQUIRED STUTTERING AND BILATERAL CEREBRAL DISEASE WILLIAM J. NOWACK University

of Arkansas

for Medical

Sciences

R. EDWARD STONE Indiana

University

School

of Medicine

Two cases of adult acquired stuttering are discussed. In both cases, the development of stuttering was associated with the appearance, in patients who had prior evidence of lefthemisphere dysfunction, of bihemispheral dysfunction. Both patients also had high anxiety levels. A course of speech therapy, which proved to be markedly anxiolytic, resulted in improvement in the communicative ability of both patients.

INTRODUCTION A syndrome of stuttering, secondary to cerebral lesions acquired in adult life, has been described by previous researchers (Canter, 1971; Helm et al., 1978; Rosenbek et al., 1978). However, the question of whether this acquired stuttering can be attributed to a specific neurological lesion or set of lesions remains unanswered, and the nature of the lesion or lesions is, as yet, unexplored (Rosenbek et al., 1978). In addition, some cases of adult acquired stuttering have been attributed to psychiatric causes (Deal, 1982). In fact, the frequency with which psychiatric disease is found in patients with stuttering has frequently, in the past, given rise to the supposition that stuttering is a hysterical manifestation (Rosenfield, 1972). The analysis of only a few cases may lead to conclusions with limited applicability. Reports of sudden-onset, adult acquired stuttering are rare (Helm et al., 1978), suggesting that the syndrome itself may be seen infrequently, although there is disagreement on that point (Rosenbek et al., 1978). The analysis of cases of such a relatively rare syndrome may be of value for understanding the role of the central nervous system in stuttering (McClean and McLean, 1985). We present two cases of adult acquired stuttering that suggest common characteristics of the underlying neurological lesion. They further suggest the possibility that the contrastSend correspondence Arkansas, for Medical

to William J. Nowack, Department of Neurology, University of Sciences, Slot #500, 4301 West Markham, Little Rock, AR 72205

0 1987 by Elsevier Science Publishing Co., Inc. 52 Vanderbilt Ave., New York, NY 10017

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142

W. J. NOWACK

ing views-that stuttering organic, nonpsychological in all cases.

DESCRIPTION

is a psychological dysfunction-may

and R. E. STONE

dysfunction or that it is an not be mutually exclusive

OF CASES

Case I In 1972 the patient, a twenty-two-year-old woman, had an episode diagnosed as viral meningitis. Other than headaches she had no sequelae. Specifically, she had no speech problem. She also had neither a past history nor a family history of speech problems. In 1976 she underwent testing for her headaches, which were thought to be depressive. Results of the neurological examination were normal, and no dysphasia, dysarthria, stuttering, or other speech problem was found. An EEG showed a brief burst of left temporal, low-voltage theta waves in waking and a similar burst during hyperventilation. Spread of the abnormal discharge to involve the right temporal area was not present. In 1979 she moved to Indiana, shortly after which, both she and her husband lost theirjobs. Considerable domestic tension ensued. She began to experience progressively severe bitemporal headaches, to have spells once or twice daily of sitting and staring blankly, and to have episodes intermittently of right-arm shaking associated with eye blinking. She developed a poorly described speech disorder and became ashamed to admit that she had been an English major in college. Neurologic examination revealed slight right-sided reflex preponderance and equivocal weakness on the right (she claimed to be right-handed). The EEG showed a left temporal spike discharge. The CT scan (without contrast) and lumbar puncture were normal. Just before cerebral angiography the patient had an episode of right-arm twitching followed by garbled speech; she then refused the procedure. For further evaluation of her progressive symptomatology she was transferred to the Indiana University Hospital. The patient’s unemployed husband stayed with her in her hospital room. He adamantly refused to consider the possibility of an emotional component to his wife’s stuttering. His solicitous and concerned behavior prolonged his wife’s hospitalization. On neurological examination, she again had signs of left-hemispheric dysfunction. The patient’s phenytoin level was found to be subtherapeutic (7.2 mcgiml). The pneumoencephalogram and radionuclide brain scan were normal. The anticonvulsant drug was stopped and, when the phenytoin level had fallen to 0. an EEG showed left anterior and midtemporal sharp waves that intermittently spread to involve the right temporal area. She stuttered on 95% of the words she spoke, repeating initial consonants four to six times and medial consonants three to four times. The

ACQUIRED STUTTERING

stuttering was not specific to any phoneme or group of phonemes. There was no adaptation effect. Whispering made no difference. She demonstrated marked effort in the production of speech but not poor eye contact, distracting sounds, or excessive body movement. Quantification of stuttering, utilizing the Riley stuttering severity instrument (Riley, 1972), resulted in a score of 23 (moderate overall stuttering). A speech pathologist felt that she had atypical stuttering, most likely on a neurogenic basis. Phenytoin, in higher doses than had been used previously, was started, and a therapeutic level (11.7 mcg/ml) was obtained. The patient was started on a progressive series of exercises consisting of nonrepetitive release of voiceless airflow in nonspeech-like tasks directed toward the production of fluent but slurred speech. She found her progress in speech therapy to be markedly anxiolytic but, nevertheless, missed several outpatient therapy appointments. Rate of articulation remained slow and articulating was imprecise. Although intelligibility was still impaired, she was stuttering only during the 10 to 15 minutes immediately following her now infrequent seizures. Although phenytoin levels were subtherapeutic (total phenytoin was 5.7 mcg/ml and free phenytoin was 0.68 mcg/ml), she decreased her phenytoin dose on her own with self-perceived speech improvement. When seen again in two months she demonstrated normal voice and speech. Speech therapy was discontinued because of her normal speech. She also became seizure-free. She progressively discontinued her anticonvulsant medication on her own without recurrence of her seizures or her dysfluency. She became reconciled with her husband and was lost to further follow-up when she moved from Indianapolis to begin a career in radio broadcasting. Case II

The patient was a 55year-old female who had experienced a left-hemisphere stroke 14 years prior to the current admission. After her first stroke she produced no language for a nine-week period and then slowly regained good functional communicative ability. Six years before the current admission she experienced a right-hemisphere cerebral infarction and was left with persistent stuttering. Left external-carotid-to-internal-carotid bypass surgery was attempted during that admission but was unsuccessful. Several years later, after a series of psychological stresses-her mother’s death and a divorce precipitated by her disability, followed shortly by her ex-husband’s suicide-she was readmitted with complaints of a worsening speech problem. She described the problem as a worsening of her dysfluency. She did not report seizure activity. Neurological examination revealed, in addition to her stuttering speech, many signs of left-hemisphere dysfunction. Some equivocal weakness and a Hoffman sign were found on the left side of the body, suggesting mild

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and R. E. STONE

right-hemisphere disease. An electroencephalogram showed a “breach” rhythm, focal slowing, and spike potentials in the left temporal area. CT scan of the head showed infarctions in the right and left hemispheres. The volume of infarcted tissue in the left hemisphere was much greater than that in the right. The phenytoin level was subtherapeutic (3.0 mcg/ml). In initial speech evaluation the patient demonstrated prolonged latency of response. However, in response to specific questioning, she stated that her hesitancies were due to stuttering and that she had no question about what words to use. She was an intelligent woman who, prior to her series of strokes, had been a research chemist. There was felt to be no reason to question her assessment of the situation. She did not stutter on final syllables, her stuttering was not phoneme bound and there was no adaptation effect. She appeared both annoyed and anxious. Her behavior reflected the tension produced by her difficulty communicating, but she did not show poor eye contact, distracting sounds, or excessive body movements. Her score on the Riley stuttering severity instrument was 3 I (severe stuttering). Dosages of the anticonvulsant, phenytoin, were adjusted to attain therapeutic levels. She was started on a treatment program consisting of modification of air flow and relaxation similar to that described in the prior case. Her overall anxiety level decreased and her stuttering improved. Rare dysphasic errors now became apparent. Because of slight ataxia and grogginess, the phenytoin level was checked and found to be slightly elevated (22.5 mcgiml). Her dosage was decreased with remission of the symptoms of toxicity. After several outpatient speech therapy sessions her score on the Riley stuttering severity instrument had improved to 20. She was pleased with her improvment.

DISCUSSION Horner and Massey have divided patients with adult acquired dysfluency into two groups: those with palilalia and those with acquired, or cortical, stuttering (ACS) (Horner and Massey, 1983). Although our two patients satisfy some criteria for both groups, they fit best into the category of ACS. Applying Canter’s classification of acquired neurogenic stuttering, our first patient can best be described as an example of dyspraxic stuttering, whereas the second patient is best described as having a mixture of dyspraxic and dysnomic stuttering (Canter, 1971). The onset of the ACS was abrupt in both patients and from neither patient, despite repeated questioning, could a history of speech problems prior to those described above be elicited. It seems unlikely, therefore, that either patient suffered from a relapse of remitted developmental stuttering. The differences between our patients and those reported in the literature reflect the well-known heterogeneity of stuttering in general (Rosenfield, 1982a) and ACS in particular.

ACQUIRED STUTTERING

145

Zimmerman and Knott (1974) found that both the right and the left inferior frontal areas performed differently in stutterers and nonstutterers. In both of our patients the ACS had its onset coincident with evidence of bihemispheral dysfunction. Other than the ACS, most of the clinical manifestations of their diseases could be explained by dysfunction of the left hemisphere alone. Although individuals can become stutterers with disease in either the right or the left hemisphere (Rosenfield, 1982b), most of the reported cases of ACS have either left-hemispheral or bihemispheral disease (Helm et al., 1978; Rosenbek et al., 1978). It has been noted that permanent ACS tends to be associated with bilateral cortical pathology and transient ACS to be associated with unilateral disease (Horner and Massey, 1983). Our first patient had bilateral hemispheral dysfunction but transient ACS. This can be interpreted to suggest that the permanence of the cortical pathology is more closely related to the permanence of the ACS than the bilaterality of the lesions. Rosenbek et al. (1978) have theorized that perhaps ACS is the result of a series of insults to the brain rather than of a single, specific cerebral lesion. Helm et al. (1978) have noted that all of their patients with stuttering coincident with cerebrovascular disease had experienced more than one discrete episode of cerebral ischemia. The temporal course of the evolution of the ACS in both of the cases is consistent with the speculation that sequential insults to the brain may be more likely to result in ACS than a comparable simultaneous bihemispheral lesion. Furthermore, the two cases suggest, but do not prove, that dysfunction in both cerebral hemispheres, with the greater clinical dysfunction in the left hemisphere, may be necessary for the development of at least some instances of ACS. Psychological disease, particularly anxiety, has been implicated in theories of stuttering (Zimmerman and Knott, 1974). Other theories have considered the etiologic role of different forms of psychopathology (Rosenfield, 1982b). Beliakova (1975) found evidence of neurotic personality disorders in over half of the population of stutterers he studied. Cases have been described in which adult acquired stuttering appeared to be unrelated to any organic central nervous system lesion and has been attributed to significant psychological distress (Deal, 1982). On the other hand, stutterers with organic disease have been dismissed as “hysterical” (Rosenlield, 1972). Zimmerman and Knott (1974) concluded from their study of the contingent negative variation that differences in interhemispheric relationships between stutterers and nonstutterers might not be a result of “anxiety.” In the first patient, development of ACS, and in the second patient, marked worsening of the ACS, were associated with both bihemispheral disease and considerable psychological stress. The psychological stress did not arise initially from the ACS, although the induced ACS soon added to the stressful situation. Furthermore, marked improvement in the ACS followed alleviation of that distress, at least in part by progress in anxiolytic speech therapy. Psychiatric intervention

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W. J. NOWACK

and R. E. STONE

may play a useful adjunctive role in the treatment of the stutterer (Rosentield, 1982b). In addition to its more traditional functions, speech therapy can also serve in the capacity of psychiatric treatment in cases where more formal intervention is felt not to be desirable. Perhaps the presence or absence of exacerbating and psychological factors can help to determine the degree to which a patient, predisposed by apparently appropriate bihemispheral cortical lesions, will experience ACS. The authors

thank

Dr. David

B. Rosenfield

Ruth Soulsby and Mrs. Bette Williams

for helpful

for assistance

criticism and suggestions and Mrs. in preparation of this manuscript.

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