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Adult onset of stuttering as a presenting sign in a parkinsonian-like syndrome: A case report
ELSEVIER
ADULT ONSET OF STUTTERING AS A PRESENTING SIGN IN A PARKINSONIAN-LIKE SYNDROME: A CASE REPORT STEVEN B. LEDER Department of Surgery, Yale University School of Medicine, New Haven. Connecticut
Stuttering onset in adulthood is rare. With no prior history of stuttering or demonstrable neurological insult, diagnosis is often that of a conversion rea,.ction. Stuttering as the first sign of a parkinsonian-like syndrome in extrapyramidal disease has only been reported once in the previous 30 years (Koller, 1983). The present case study differs from and builds upon the case reports in the literature, and describes a 29-year-old white male who began stuttering purportedly secondary to psychological stress. The fluency evaluation revealed severe stuttering characterized by multiple repetitions and/or blocks, with 20 or more repetitions per word routinely noted during both conversational speech and oral reading. No starters or secondary stuttering characteristics, no specific word fears or avoidances, and no situational fears were exhibited. The subsequent neurological examination found resting tremor in hands and legs, lingual fasciculations, gait imbalance, and numbness and tingling of the hands and feet, all of which were progressive in nature. Rehabilitation initially focused on fluency therapy, but then included psychiatric therapy, and finally medical intervention. Fluency and psychiatric therapies were unsuccessful in eliminating stuttering. Following a diagnosis of parkinsonian-like syndrome, medical intervention with carbidopa-levodopa resulted in dramatic improvement of motor, sensory, and fluency symptoms. It is important to consider extrapyramidal disease as an etiological factor in patients with adult onset of stuttering. © 1996 by Elsevier Science Inc. Educational Objectives: The reader will (1) understand the three general etiologies for adult onset of stuttering; (2) acquire knowledge and understanding of elements characteristic of stuttering subsequent to psychological stress; and (3) develop knowledge regarding the diffewrential diagnosis of stuttering as a conversation reaction versus a parkinsonian-like syndrome.
INTRODUCTION Stuttering onset in adulthood is rare (Deal, 1982), and should be investigated carefully (Van Riper, 1972). There appear to be three general etiologies for
Address correspondence to Steven B. Leder, Ph.D.. Department of Surgery, Section of Otolaryngology, Yale University School of Medicine, 20 York St, YPB-468, New Haven. CT 06504. J. COMMUN. DISORD. 29 (t996), 471-478 © 1996 by Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
0021-9924/96/$ 15.00 SSDI 0021-9924(95)00055-I
~2
LEDER
adult onset of stuttering: (1) neurological insult, i.e., cerebrovascular accident or trauma (Canter, 1971; Helm, Butler, & Benson, 1978; Rosenbek, Messert, Collins, & Wertz, 1978; Rosenfield, 1972); (2) stutterers who have relapses or interiorized their stuttering and are unable to control it under increased stress (Van Riper, 1971); and (3) conversion reactions in the absence of prior history of stuttering or demonstrable neurological insult (Deal, 1982). The literature contains four case reports that describe stuttering as a conversion reaction. The first case report (Wallen, 1971) discussed adult onset of stuttering following acute psychological stress in a previously well functioning and successful individual. The precipitating cause of stuttering in the second case was psychological trauma caused by an explosion (Dempsey & Granich, 1978). In the third case report, Deal (1982) described an individual who exhibited a significant and long-standing psychiatric history of which chronic stuttering was one of many problems. Deal and Doro (1987) described a fourth case in which stuttering was episodic in nature. The present case study reports adult onset of stuttering as the first sign of a parkinsonian-like syndrome in extrapyramidal disease. This case is noteworthy as a literature review over the past 30 years revealed only one report dealing with stuttering as an early sign in parkinsonism (Koller, 1983).
Case Report Mr. A., a 29-year-old white male, was referred initially to speech pathology by his primary care physician following an acute onset of dysfluent speech. There was no reported history of stuttering prior to the current onset. Mr. A. is a graphic designer at a university, and has been under stress during the last few months while working on a museum catalog. His only medical complaint was of headaches, for which he took aspirin as needed. On November 21, 1993, the first episode of stuttering occurred on the word, "paper." Dysfluencies increased during the morning of the following day, and severe dysfluency was exhibited by early afternoon. He reported that his vocal pitch was normal at that time. Pitch became higher over the next three to four days before returning to normal over the next month, but severe dysfluency persisted. The only other problem noted was an inability to write in capital letters; lower case printing and cursive writing were unimpaired. (In his work as a graphic designer he only used capital letters. It is also interesting to note that his first dysfluent word was "paper," a material necessary for his job.) The writing difficulty lasted seven days and then resolved spontaneously. The above presenting history appeared consistent with a diagnosis of conversion reaction. Neurologic diagnostic evaluations in November, 1993 were normal, i.e., EEG, MRI without gadolinium and CT scans of the brain, and lumbar punc-
ADULT ONSET OF STUTTERING
473
ture. Except for his father, age 58, who has had complex partial seizures since his early 20s, there was no family history of neurological disease.
Speech Pathology Mr. A. was evaluated on December 9, 1993, 19 days following onset of stuttering. He presented with severe dysfluency characterized by frequent syllable and word repetitions, with infrequent blocks on syllables and words. Every syllable and word exhibited multiple repetitions and/or blocks, with 20 or more repetitions per syllable routinely noted during both conversational speech and oral reading. Choral reading did not alter the stuttering pattern. Also, like Deal's (1982) case report, stuttering was exhibited during conversational speech and reading when mouthing words without voicing. No receptive or expressive language or cognitive function changes were reported. In addition, vocal production was characterized by increased pitch secondary to observational tension in the shoulder and neck areas. No starters or secondary characteristics, no specific word fears or avoidances, and no situational fears, e.g., telephone use, were exhibited. There were no limb tremors or lingual fasciculations at this time. Mr. A. was very distressed about his stuttering and was motivated to correct it. Based on history and fluency evaluation, a working diagnosis was made of adult onset of stuttering secondary to conversion reaction.
Fluency Therapy Fluency therapy occurred three times a week. Specific therapy techniques included: 1. Continuous breath flow to promote an open glottis followed by working from a whisper and then progressing to voicing by humming vowels and nasal continuants/re,n/; 2. Soft contacts on stop plosive and fricative phonemes: 3. Relaxation of the shoulders and neck area. Mr. A. was able to understand and incorporate the above techniques adequately during the initial session to begin to control his stuttering and to increase his ability to produce fluent utterances. A discussion focusing on responsibility for and control of Mr. A.'s stuttering followed the initial success of fluency therapy. Using breath flow. soft contacts, and negative practice Mr. A. demonstrated self-control of both stuttered and fluent speech production. Hierarchical fluency shaping goals were delineated: 1. Using breath flow, soft contacts, and relaxation start with rote speech
474
LEDER
tasks, e.g., counting and days of the week, and short, overlearned phrases, e.g., How are you? and Good-bye, progressing to conversational speech as quickly as possible while maintaining fluency; 2. Increase rate of speech while maintaining fluency; 3. Increase loudness of speech while maintaining fluency; 4. Maintain appropriate vocal pitch by decreasing tension in the shoulders and neck. The above goals were pursued for three months. In addition, daily telephone contact occurred to aid in maintenance, carry-over, and generalization to conversational speech. However, except for pitch returning to normal, Mr. A. did not continue to exhibit improvement. In March, 1994 dysfluent speech became more severe and other motor anomalies, e.g., lingual fasciculations and hand and leg tremors, were observed. A referral for further neurological evaluation was made.
Psychiatry Psychiatric intervention began in December, 1993, based upon a diagnosis of a conversion reaction. Doxepin hydrochloride, 200 mg at night, was prescribed for depression and sleep. The psychiatrist and speech-language pathologist conferred by telephone biweekly to discuss behavioral and psychiatric strategies to improve Mr. A.'s stuttering. There was, however, no improvement in Mr. A.'s fluency secondary to the psychiatric intervention, and by March, 1994 the psychiatrist felt the stuttering was of organic origin.
Neurology A second neurological evaluation was conducted on August 23, 1994. SPECT scanning showed normal proportions of dopamine which raised the possibility that the basis for Mr. A.'s disability was either receptor dysfunction or no biochemical basis for the symptoms. Cranial nerve examination was normal except for markedly slow tongue movements. Sensory examination was normal to all primary and cortical modalities. Cerebellar examination showed normal finger to nose movements in procession, but movements were slow. Tremor persisted during these movements but did not increase in amplitude. Motor examination showed a tremor at rest in both arms, right more than left, and a slight tremor at rest in the right leg. He was able to stand but not walk on his heels and toes. Balance was judged to be slightly abnormal because pushing on his chest and back produced mild instability with no reflexive compensatory movements of his arms. His face was without expression. All movements were slow to begin, and once started most movements were slow to complete. He was unable to perform rapid alternating movements, e.g., clap or hit the
ADULT ONSET OF STUTTERING
475
back of his hand to the other hand, or arise from a chair without the use of his hands. Although Mr. A.'s clinical syndrome was difficult to characterize, the vast majority of clinical features were most suggestive of parkinsonism. Carbidopa-levodopa (25-100) was prescribed. The initial dose was four times a day (August, 1994), with reduction to three times a day (February, 1995), two times a day (April, 1995), and one time a day (May, 1995). Dose reductions occurred due to improvements in both speech and motor skills. Mr. A. was sensitive to the dose, since during peak effectiveness his fluency, fine motor skills, and fluidity of movement improved. He was able to speak in short, i.e., three to five word, utterances without stuttering. However, as the dose was decreased stuttering was observed to increase in severity. Following the diagnosis of a parkinsonian-like syndrome in August, 1994, psychiatric intervention focused on support and coping with a disease process. Mr. A. reported positive psychological gains during this very upsetting time in his life.
DISCUSSION Mr. A. presented initially with a majority of elements characteristic of stuttering subsequent to psychological stress. Specifically: (1) onset was sudden; (2) onset was related temporally to psychological pressure; (3) stuttering was primarily characterized by repetitions of initial or stressed syllables; (4) stuttering was not affected by communicative situations; (5) all types of speech were stuttered equally; and (6) no starters, secondary characteristics, word fears, word avoidances, or situational fears were exhibited (Deal, 1982). However, Mr. A.'s stuttering did not respond to either fluency or psychiatric therapy, and as his stuttering worsened other motor abnormalities appeared. It was not until treatment with carbidopa-levodopa was initiated that Mr. A.'s stuttering improved to the point where conversational speech could be maintained with minimal dysfluencies. Although a conversion reaction cannot absolutely be eliminated as an etiology of Mr. A.'s stuttering, the more probable explanation is a parkinsonian-like syndrome based upon observed neurological abnormalities and response to medication. Although it is well known that extrapyramidal disease causes specific speech disturbances, i.e., monotony of pitch and loudness, reduced stress, short phrases, variable rate, short rushes of speech, and imprecise consonants (Critchley, 1981; Darley, Aronson, and Brown, 1975), stuttering as a sign of extrapyramidal disease has not been well documented. In the only other report in the literature to date, Koller (1983) described six patients with extrapyramidal disease who exhibited stuttering. Stuttering in extrapyramidal disease differs from stuttering secondary to vascular disease or head trauma due to the rare occurrence of dysfluencies during choral and repetitive speech, a positive adaptation effect, and lack of language impairment. Also, stuttering in ex-
476
LEDER
trapyramidal disease differs from developmental stuttering in that there are no secondary motor characteristics or behavioral changes associated with stuttering, and stuttering is present during singing (Koller, 1983). Mr. A.'s stuttering was similar to the above characteristics except that he did not exhibit an adaptation effect, and his stuttering persisted during choral and repetitive speech. Perhaps the large age difference between Koller's (1983) patients, mean age 61 years versus 29 years for Mr. A., accounted for the differences in stuttering characteristics observed. Although Koller (1983) reported that stuttering occurred early in the course of the disease, only one case exhibited stuttering as the initially observed sign. The present report describes the second case of stuttering as the presenting sign of extrapyramidal disease, and highlights the importance of considering extrapyramidal disease when acute onset of adult stuttering is observed, especially in the younger adult. It is hoped that this case stimulates further discussion concerning the wide range of factors that cause stuttering. The author thanks Drs. J. F. Kveton and B. C. Watson for insightful comments on an earlier version of the manuscript.
REFERENCES Canter, G.J. (1971). Observations on neurogenic stuttering: A contribution to differential diagnosis. British Journal of Disorders of Communication 6, 139-143. Critchley, E.M.R. (1981). Speech disorders of parkinsonism: A review. Journal of Neurology, Neurosurgery, and Psychiatry 44, 751-758. Darley, D.A., Aronson, A.E., & Brown, J.R. (1975). Motor speech disorders. Philadelphia: W.B. Saunders Co. Deal, J.L. (1982). Sudden onset of stuttering: a case report. Journal of Speech and Hearing Disorders 47, 301-304. Deal, J.L., & Doro, J.M. (1987). Episodic hysterical stuttering. Journal of Speech and Hearing Disorders 52, 299-300. Dempsey, G.L., & Granich, M. (1978). Hypno-behavioral therapy in the case of a traumatic stutterer: a case study. Journal of Clinical and Experimental Hypnosis 26, 125-133. Helm, N.A., Butler, R.B., & Benson, D.F. (1978). Acquired stuttering. Neurology28, 1159-1165. Koller, W.C. (1983). Dysfluency (stuttering) in extrapyramidal disease. Archives of Neurology 40, 175-177.
ADULT ONSET OF STUTI'ERING
477
Rosenbek, J., Messert, B., Collins, M., & Wertz, R.T. (1978). Stuttering following brain damage. Brain and Language 6, 82-96. Rosenfield, D.B. (1972). Stuttering and cerebral ischemia (letter to the editor). New England Journal of Medicine 287, 991. Van Riper, C. (1971). The nature of stuttering. Englewood-Cliffs, NJ: Prentice-Hall, Inc. Wallen, V. (1961). Primary stuttering in a 28-year-old adult. Journal of Speech and Hearing Disorders 26, 394--395. Manuscript received April 17, 1995; revised July 20, 1995.
CONTINUING EDUCATION
Adult Onset of Stuttering as a Presenting Sign in a Parkinsonian-Like Syndrome: Case Report QUESTIONS 1. Stuttering onset in adulthood is: a. Common b. Episodic c. Rare d. Only due to neurological insult e. Easily treatable 2. Which is not an etiology of stuttering onset in adulthood?: a. Cerebrovascular accident b. Stuttering relapses c. Acute psychological stress d. Senility e. Traumatic brain injury 3. A conversion reaction is when: a. There is no prior history of stuttering or demonstrable neurological insult b. Stuttering is very severe c. A stuttering relapse occurrs d. Stuttering changes from repetitions to prolongations e. Stuttering is eliminated 4. Stuttering characteristics that should alert you to a parkinsonian-like syndrome are: a. Specific word fears
478
b. c. d. e.
LEDER
Progressive motor abnormalities Secondary stuttering characteristics Situation fears Characteristic starters
5. Extrapyramidal disease causes specific speech disturbances except: a. Reduced stress b. Variable rate c. Short phrases d. Monotony of pitch and loudness e. Strained strangled voice
ADULT ONSET OF STUTTERING AS A PRESENTING SIGN IN A PARKINSONIAN-LIKE SYNDROME: A CASE REPORT STEVEN B. LEDER Department of Surgery, Yale University School of Medicine, New Haven. Connecticut
Stuttering onset in adulthood is rare. With no prior history of stuttering or demonstrable neurological insult, diagnosis is often that of a conversion rea,.ction. Stuttering as the first sign of a parkinsonian-like syndrome in extrapyramidal disease has only been reported once in the previous 30 years (Koller, 1983). The present case study differs from and builds upon the case reports in the literature, and describes a 29-year-old white male who began stuttering purportedly secondary to psychological stress. The fluency evaluation revealed severe stuttering characterized by multiple repetitions and/or blocks, with 20 or more repetitions per word routinely noted during both conversational speech and oral reading. No starters or secondary stuttering characteristics, no specific word fears or avoidances, and no situational fears were exhibited. The subsequent neurological examination found resting tremor in hands and legs, lingual fasciculations, gait imbalance, and numbness and tingling of the hands and feet, all of which were progressive in nature. Rehabilitation initially focused on fluency therapy, but then included psychiatric therapy, and finally medical intervention. Fluency and psychiatric therapies were unsuccessful in eliminating stuttering. Following a diagnosis of parkinsonian-like syndrome, medical intervention with carbidopa-levodopa resulted in dramatic improvement of motor, sensory, and fluency symptoms. It is important to consider extrapyramidal disease as an etiological factor in patients with adult onset of stuttering. © 1996 by Elsevier Science Inc. Educational Objectives: The reader will (1) understand the three general etiologies for adult onset of stuttering; (2) acquire knowledge and understanding of elements characteristic of stuttering subsequent to psychological stress; and (3) develop knowledge regarding the diffewrential diagnosis of stuttering as a conversation reaction versus a parkinsonian-like syndrome.
INTRODUCTION Stuttering onset in adulthood is rare (Deal, 1982), and should be investigated carefully (Van Riper, 1972). There appear to be three general etiologies for
Address correspondence to Steven B. Leder, Ph.D.. Department of Surgery, Section of Otolaryngology, Yale University School of Medicine, 20 York St, YPB-468, New Haven. CT 06504. J. COMMUN. DISORD. 29 (t996), 471-478 © 1996 by Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
0021-9924/96/$ 15.00 SSDI 0021-9924(95)00055-I
~2
LEDER
adult onset of stuttering: (1) neurological insult, i.e., cerebrovascular accident or trauma (Canter, 1971; Helm, Butler, & Benson, 1978; Rosenbek, Messert, Collins, & Wertz, 1978; Rosenfield, 1972); (2) stutterers who have relapses or interiorized their stuttering and are unable to control it under increased stress (Van Riper, 1971); and (3) conversion reactions in the absence of prior history of stuttering or demonstrable neurological insult (Deal, 1982). The literature contains four case reports that describe stuttering as a conversion reaction. The first case report (Wallen, 1971) discussed adult onset of stuttering following acute psychological stress in a previously well functioning and successful individual. The precipitating cause of stuttering in the second case was psychological trauma caused by an explosion (Dempsey & Granich, 1978). In the third case report, Deal (1982) described an individual who exhibited a significant and long-standing psychiatric history of which chronic stuttering was one of many problems. Deal and Doro (1987) described a fourth case in which stuttering was episodic in nature. The present case study reports adult onset of stuttering as the first sign of a parkinsonian-like syndrome in extrapyramidal disease. This case is noteworthy as a literature review over the past 30 years revealed only one report dealing with stuttering as an early sign in parkinsonism (Koller, 1983).
Case Report Mr. A., a 29-year-old white male, was referred initially to speech pathology by his primary care physician following an acute onset of dysfluent speech. There was no reported history of stuttering prior to the current onset. Mr. A. is a graphic designer at a university, and has been under stress during the last few months while working on a museum catalog. His only medical complaint was of headaches, for which he took aspirin as needed. On November 21, 1993, the first episode of stuttering occurred on the word, "paper." Dysfluencies increased during the morning of the following day, and severe dysfluency was exhibited by early afternoon. He reported that his vocal pitch was normal at that time. Pitch became higher over the next three to four days before returning to normal over the next month, but severe dysfluency persisted. The only other problem noted was an inability to write in capital letters; lower case printing and cursive writing were unimpaired. (In his work as a graphic designer he only used capital letters. It is also interesting to note that his first dysfluent word was "paper," a material necessary for his job.) The writing difficulty lasted seven days and then resolved spontaneously. The above presenting history appeared consistent with a diagnosis of conversion reaction. Neurologic diagnostic evaluations in November, 1993 were normal, i.e., EEG, MRI without gadolinium and CT scans of the brain, and lumbar punc-
ADULT ONSET OF STUTTERING
473
ture. Except for his father, age 58, who has had complex partial seizures since his early 20s, there was no family history of neurological disease.
Speech Pathology Mr. A. was evaluated on December 9, 1993, 19 days following onset of stuttering. He presented with severe dysfluency characterized by frequent syllable and word repetitions, with infrequent blocks on syllables and words. Every syllable and word exhibited multiple repetitions and/or blocks, with 20 or more repetitions per syllable routinely noted during both conversational speech and oral reading. Choral reading did not alter the stuttering pattern. Also, like Deal's (1982) case report, stuttering was exhibited during conversational speech and reading when mouthing words without voicing. No receptive or expressive language or cognitive function changes were reported. In addition, vocal production was characterized by increased pitch secondary to observational tension in the shoulder and neck areas. No starters or secondary characteristics, no specific word fears or avoidances, and no situational fears, e.g., telephone use, were exhibited. There were no limb tremors or lingual fasciculations at this time. Mr. A. was very distressed about his stuttering and was motivated to correct it. Based on history and fluency evaluation, a working diagnosis was made of adult onset of stuttering secondary to conversion reaction.
Fluency Therapy Fluency therapy occurred three times a week. Specific therapy techniques included: 1. Continuous breath flow to promote an open glottis followed by working from a whisper and then progressing to voicing by humming vowels and nasal continuants/re,n/; 2. Soft contacts on stop plosive and fricative phonemes: 3. Relaxation of the shoulders and neck area. Mr. A. was able to understand and incorporate the above techniques adequately during the initial session to begin to control his stuttering and to increase his ability to produce fluent utterances. A discussion focusing on responsibility for and control of Mr. A.'s stuttering followed the initial success of fluency therapy. Using breath flow. soft contacts, and negative practice Mr. A. demonstrated self-control of both stuttered and fluent speech production. Hierarchical fluency shaping goals were delineated: 1. Using breath flow, soft contacts, and relaxation start with rote speech
474
LEDER
tasks, e.g., counting and days of the week, and short, overlearned phrases, e.g., How are you? and Good-bye, progressing to conversational speech as quickly as possible while maintaining fluency; 2. Increase rate of speech while maintaining fluency; 3. Increase loudness of speech while maintaining fluency; 4. Maintain appropriate vocal pitch by decreasing tension in the shoulders and neck. The above goals were pursued for three months. In addition, daily telephone contact occurred to aid in maintenance, carry-over, and generalization to conversational speech. However, except for pitch returning to normal, Mr. A. did not continue to exhibit improvement. In March, 1994 dysfluent speech became more severe and other motor anomalies, e.g., lingual fasciculations and hand and leg tremors, were observed. A referral for further neurological evaluation was made.
Psychiatry Psychiatric intervention began in December, 1993, based upon a diagnosis of a conversion reaction. Doxepin hydrochloride, 200 mg at night, was prescribed for depression and sleep. The psychiatrist and speech-language pathologist conferred by telephone biweekly to discuss behavioral and psychiatric strategies to improve Mr. A.'s stuttering. There was, however, no improvement in Mr. A.'s fluency secondary to the psychiatric intervention, and by March, 1994 the psychiatrist felt the stuttering was of organic origin.
Neurology A second neurological evaluation was conducted on August 23, 1994. SPECT scanning showed normal proportions of dopamine which raised the possibility that the basis for Mr. A.'s disability was either receptor dysfunction or no biochemical basis for the symptoms. Cranial nerve examination was normal except for markedly slow tongue movements. Sensory examination was normal to all primary and cortical modalities. Cerebellar examination showed normal finger to nose movements in procession, but movements were slow. Tremor persisted during these movements but did not increase in amplitude. Motor examination showed a tremor at rest in both arms, right more than left, and a slight tremor at rest in the right leg. He was able to stand but not walk on his heels and toes. Balance was judged to be slightly abnormal because pushing on his chest and back produced mild instability with no reflexive compensatory movements of his arms. His face was without expression. All movements were slow to begin, and once started most movements were slow to complete. He was unable to perform rapid alternating movements, e.g., clap or hit the
ADULT ONSET OF STUTTERING
475
back of his hand to the other hand, or arise from a chair without the use of his hands. Although Mr. A.'s clinical syndrome was difficult to characterize, the vast majority of clinical features were most suggestive of parkinsonism. Carbidopa-levodopa (25-100) was prescribed. The initial dose was four times a day (August, 1994), with reduction to three times a day (February, 1995), two times a day (April, 1995), and one time a day (May, 1995). Dose reductions occurred due to improvements in both speech and motor skills. Mr. A. was sensitive to the dose, since during peak effectiveness his fluency, fine motor skills, and fluidity of movement improved. He was able to speak in short, i.e., three to five word, utterances without stuttering. However, as the dose was decreased stuttering was observed to increase in severity. Following the diagnosis of a parkinsonian-like syndrome in August, 1994, psychiatric intervention focused on support and coping with a disease process. Mr. A. reported positive psychological gains during this very upsetting time in his life.
DISCUSSION Mr. A. presented initially with a majority of elements characteristic of stuttering subsequent to psychological stress. Specifically: (1) onset was sudden; (2) onset was related temporally to psychological pressure; (3) stuttering was primarily characterized by repetitions of initial or stressed syllables; (4) stuttering was not affected by communicative situations; (5) all types of speech were stuttered equally; and (6) no starters, secondary characteristics, word fears, word avoidances, or situational fears were exhibited (Deal, 1982). However, Mr. A.'s stuttering did not respond to either fluency or psychiatric therapy, and as his stuttering worsened other motor abnormalities appeared. It was not until treatment with carbidopa-levodopa was initiated that Mr. A.'s stuttering improved to the point where conversational speech could be maintained with minimal dysfluencies. Although a conversion reaction cannot absolutely be eliminated as an etiology of Mr. A.'s stuttering, the more probable explanation is a parkinsonian-like syndrome based upon observed neurological abnormalities and response to medication. Although it is well known that extrapyramidal disease causes specific speech disturbances, i.e., monotony of pitch and loudness, reduced stress, short phrases, variable rate, short rushes of speech, and imprecise consonants (Critchley, 1981; Darley, Aronson, and Brown, 1975), stuttering as a sign of extrapyramidal disease has not been well documented. In the only other report in the literature to date, Koller (1983) described six patients with extrapyramidal disease who exhibited stuttering. Stuttering in extrapyramidal disease differs from stuttering secondary to vascular disease or head trauma due to the rare occurrence of dysfluencies during choral and repetitive speech, a positive adaptation effect, and lack of language impairment. Also, stuttering in ex-
476
LEDER
trapyramidal disease differs from developmental stuttering in that there are no secondary motor characteristics or behavioral changes associated with stuttering, and stuttering is present during singing (Koller, 1983). Mr. A.'s stuttering was similar to the above characteristics except that he did not exhibit an adaptation effect, and his stuttering persisted during choral and repetitive speech. Perhaps the large age difference between Koller's (1983) patients, mean age 61 years versus 29 years for Mr. A., accounted for the differences in stuttering characteristics observed. Although Koller (1983) reported that stuttering occurred early in the course of the disease, only one case exhibited stuttering as the initially observed sign. The present report describes the second case of stuttering as the presenting sign of extrapyramidal disease, and highlights the importance of considering extrapyramidal disease when acute onset of adult stuttering is observed, especially in the younger adult. It is hoped that this case stimulates further discussion concerning the wide range of factors that cause stuttering. The author thanks Drs. J. F. Kveton and B. C. Watson for insightful comments on an earlier version of the manuscript.
REFERENCES Canter, G.J. (1971). Observations on neurogenic stuttering: A contribution to differential diagnosis. British Journal of Disorders of Communication 6, 139-143. Critchley, E.M.R. (1981). Speech disorders of parkinsonism: A review. Journal of Neurology, Neurosurgery, and Psychiatry 44, 751-758. Darley, D.A., Aronson, A.E., & Brown, J.R. (1975). Motor speech disorders. Philadelphia: W.B. Saunders Co. Deal, J.L. (1982). Sudden onset of stuttering: a case report. Journal of Speech and Hearing Disorders 47, 301-304. Deal, J.L., & Doro, J.M. (1987). Episodic hysterical stuttering. Journal of Speech and Hearing Disorders 52, 299-300. Dempsey, G.L., & Granich, M. (1978). Hypno-behavioral therapy in the case of a traumatic stutterer: a case study. Journal of Clinical and Experimental Hypnosis 26, 125-133. Helm, N.A., Butler, R.B., & Benson, D.F. (1978). Acquired stuttering. Neurology28, 1159-1165. Koller, W.C. (1983). Dysfluency (stuttering) in extrapyramidal disease. Archives of Neurology 40, 175-177.
ADULT ONSET OF STUTI'ERING
477
Rosenbek, J., Messert, B., Collins, M., & Wertz, R.T. (1978). Stuttering following brain damage. Brain and Language 6, 82-96. Rosenfield, D.B. (1972). Stuttering and cerebral ischemia (letter to the editor). New England Journal of Medicine 287, 991. Van Riper, C. (1971). The nature of stuttering. Englewood-Cliffs, NJ: Prentice-Hall, Inc. Wallen, V. (1961). Primary stuttering in a 28-year-old adult. Journal of Speech and Hearing Disorders 26, 394--395. Manuscript received April 17, 1995; revised July 20, 1995.
CONTINUING EDUCATION
Adult Onset of Stuttering as a Presenting Sign in a Parkinsonian-Like Syndrome: Case Report QUESTIONS 1. Stuttering onset in adulthood is: a. Common b. Episodic c. Rare d. Only due to neurological insult e. Easily treatable 2. Which is not an etiology of stuttering onset in adulthood?: a. Cerebrovascular accident b. Stuttering relapses c. Acute psychological stress d. Senility e. Traumatic brain injury 3. A conversion reaction is when: a. There is no prior history of stuttering or demonstrable neurological insult b. Stuttering is very severe c. A stuttering relapse occurrs d. Stuttering changes from repetitions to prolongations e. Stuttering is eliminated 4. Stuttering characteristics that should alert you to a parkinsonian-like syndrome are: a. Specific word fears
478
b. c. d. e.
LEDER
Progressive motor abnormalities Secondary stuttering characteristics Situation fears Characteristic starters
5. Extrapyramidal disease causes specific speech disturbances except: a. Reduced stress b. Variable rate c. Short phrases d. Monotony of pitch and loudness e. Strained strangled voice