Neurologic manifestations of childhood hysteria

Neurologic manifestations of childhood hysteria

B E H A V I O R A L P E D I A T R I C S Richard W. Olmsted, E d i t o r Neurologic manifestations of childhood hysteria During a four and one-half-...

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B E H A V I O R A L

P E D I A T R I C S

Richard W. Olmsted, E d i t o r

Neurologic manifestations of childhood hysteria During a four and one-half-year period, 32 children (23 girls, 9 bo.vs) referred for neurologie evahtation were diagnosed as having conversion hysteria. These patients were reviewed to develop a composite picture of the neurologic s)'mptoms o f childhood hysteria. Presenting complaints inchtded seizttres, para!vsis, syncope, vertigo, pain, paresthesias, blindness, diplopia, blurred vision, tics, and intellectual deterioration. TwenO'-four patients required hospitalization due to the acuteness o f their presenting ,~ivmptoms: the average hospital stay was six days. Twenty percent of the hospitali-ed patients remitted spontaneousl),, but 50% required extensive ps)'chiatric evaluation and therapy before intprovement. Di[ficul~v Of diagnosis was most apparent in the pseudoseizure population, all of whom could readih' mimic seizure activity and most of whom were receiving anticonvulsants. A n intportant differential feature in the psendoseizure group was the usual normalcy o f the electroencephalogram. In none of the patients was hysteria considered by the referring physician.

Sanford Schneider, M.D.,* L o m a

Linda,

Calif., and David R. Rice, M . D . ,

Boston, Mass.

FREUD in 1893 called hysteria the most enigmatic of all nervous disorders, and recognized that hysteria was an important part of the differential diagnosis in many patients with hemianesthesia, visual field restriction and epileptiform convulsions. 1 Hysteria masquerading as a seizure disorder in childhood was first reported in America in 1668, when the four children of John Goodwin of Boston, "remarkable for their piety, honesty and industry," were taken with "odd" fits which were attributed to witchcraft.'-' Sheffield in 1898 was probably the first American to review hysteria in childhood comprehensively.:' His report incorporated the pioneering studies of Abraham Jacobi, S. Weir Mitchell, and Bernard Sachs, who recognized the frequency of convulsions and blindness as manifestations of childhood hysteria. Pediatric texts at the turn of the century tended to devote chapters to hysteria,' whereas modern texts have curtailed their discussions? A review of our patients seen over a 4~A-year period of time indicated that nearly 2% of our pediatric

neurology inpatient population was ultimately diagnosed as having, conversion hysteria, suggesting that the diagnosis of hysteria is far more common than generally appreciated. Our data were analyzed to develop a profile of hysterical children who present with neurologic symptoms.

MATERIALS AND METHODS The records of 32 children seen between September, 1971, and June, 1976, with a diagnosis of conversion hysteria were reviewed. All patients were referred by pediatricians, general practitioners, or neurologists. Over 20 additional patients with suspected psychogenic headaches were excluded from the study because of the difficulty &objective evaluation. The diagnosis of conversion hysteria was based on a combination of the clinical features, physical, and neurologic findings and the subsequent course.

RESULTS From Loma Linda University School o f Medicine and Tufts University School o f Medicine. Presented in part to the anmtal meeting o f the Child Neurology Society, Monterel,, California, October 29, 1976. *Reprint address: Division of Child Neurology, Room A-531, Loma Linda University Medical Center. Loma Lin&z, CA 92350.

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Our patients varied from 6 to 18 years, with a mean of 13 years. The female to male ratio was 2 to 1, but the incidence of conversion hysteria between the sexes was approximately equal below the age of 10. The major presenting symptoms, in order of frequency, included seizures (8), paralysis (6), syncope (4), pain/paresthesias (4), dizziness (4), visual loss (2), tics (2), diplopia (1), and The Journal o f P E D I A T R I C S Vol. 94, No. 1, pp. 153-156

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intellectual deterioration (l). With the exception of one patient who had a tic, all presented with numerous polysymptomatic secondary complaints including the nearly universal complaint of headache, which was recorded in 83% of the female population. This percentage is similar to the percentage found in an adult female hysterical population." In the paralytic groups, there was very little clustering in any given sex or age range. Syncope was observed only in girls. In the eight patients with pseudoseizures, the six girls ranged from 11 to 18 years with most patients in the mid-to-latter ten years: the two boys were 13 and 14. A marked difference was noted between the two boys and six girls as to the duration of seizure activity. In girls, chronicity of seizures was common, exceeding one year in 50%; additionally, seizure frequency was more frequent in females, tending to occur daily. Electroencephalograms in patients with pseudoseizures were normal except for a minor abnormality in one patient. Seventy percent of our patients required an average of six days for hospitalization, either due to the acuteness of the presenting complaint or at the referring physician's request. During this period of hospitalization, 20% remitted spontaneously; the remainder required psychiatric therapy. All of the inpatients, except for one, resolved within three weeks after the diagnosis of conversion hysteria was made and appropriate psychiatric treatment was initiated, including those patients who had had symptoms for more than three years. The one patient who continued to have seizures after a diagnosis of conversion hysteria was established was shown to have a persistent adjustment reaction of childhood with psychotic features. Social Service intake evaluation revealed that nearly all the patients with conversion hysteria had a marked alteration of family dynamics. In most families the interpersonal difficulties were chronic; seldom was the hysteria related to acute situational reactions. Ninety percent of our patients were first or second in the birth order, which may be of etiologic importance in the pathogenesis of a hysterical personality. Family size seemed to be of little etiologic importance. All patients who had formal psychometric testing (nearly all the inpatients) had significant hysterial features on either the Thematic or Childhood Apperception tests or the Minnesota Multiphasic Personality Inventory. Eight patients (seven of eight being outpatients) were treated without formal psychiatric intervention by informal counseling, social service intervention, and medication (chlordiazepoxide or thioridazine) to relieve anxiety. Medication was always given with a strong positive suggestion that resolution of the problem would occur immediately. In none of these patients did the presenting

The Journal of Pediatrics January 1979

neurologic symptoms recur as long as the patient was being followed by the neurologist. However, 50% of the outpatient population has been lost to follow-up and it is unknown whether further hysterical symptoms subsequently developed. CASE REPORTS Patient 1. A 12-year-old girl, referred by a neurologist, had a one-year history of generalized seizure activity. At the time of referral, she was maintained on multiple anticonvulsant drugs and having daily, well-described generalized seizures. Past history was unremarkable except for a strong family history of migraine. Results of general physical and neurologic examinations were normal. The patient was hospitalized and routine studies were all normal except for a mildly paroxysmal electroencephalogram. Despite numerous changes in anticonvulsant medications, at times producing almost toxic serum levels, she continued to have generalized seizures for 6 months. Her seizures were never observed by the professional staff and, despite many falls, she was never noted to be bruised. Two follow-up electroencephalograms were unchanged. On an outpatient visit, she complained of diplopia, which was clearly hysterical on red glass testing. At this time a marked sibling conflict with a highly achieving older sister was uncovered and, two weeks after the initiation of outpatient child psychiatry visits, her seizures resolved and anticonvulsant therapy was discontinued. Patient 2. A 16-year-old girl with a well-described and observed left focal seizure disorder had been maintained without success on many anticonvulsant drugs for one year. Physical and neurologic examinations were unremarkable; her electroencephalogram was normal. During her initial evaluation, cerebral angiography showed a questionable early draining vein, raising the suspicion of a low-grade glioma. She was then maintained on various anticonvulsant medications for a second year without seizure control. At this time her father's anxiety and our inability to control his daughter's seizure activity led to individual questioning of the girl. At this time, marked abnormalities in the family dynamics, including the father's incestuous relationship with her, became known. After the child's removal from the home, her seizure activity cleared dramatically and all medications were discontinued.

DISCUSSION During the years covered by this study, no referring physician ever suggested that conversion hysteria was possibly producing neurologic symptoms, indicating that the neurologic symptoms of conversion hysteria are infrequently recognized by the family physician and pediatrician. This surprisingly large n u m b e r of children would cause us to speculate that some children carried with suspected chronic neurologic disease, especially seizures, may in reality have entrenched conversion hysteria. Robbins and O'NeaF reported only 41 cases of hysteria among 51,000 patients admitted to St. Louis Children's Hospital over a 15-year period. Stevens" stated that only

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39 children evaluated over 12 years were discharged from D. C. Children's Hospital with a diagnosis of hysteria. Lewis~'reported that only five patients in 129 requests for child psychiatric consultation in both in- and outpatients had a diagnosis of conversion hysteria. Our data would imply that conversion reaction is far more frequent and often overlooked in the differential diagnosis of neurologic symptoms, as pointed out by Jensen and Weft.'<' Pseudoseizures constitute the largest number of patients in this series and represented the most vexing difficulty in diagnosis. A pseudoseizure, well described by the parents, represents an authentic seizure until witnessed by the physician or until examination reveals hysterical features which raises doubt as to the diagnosis. Two of our patients were felt to have valid seizure disorders for six to 12 months before a diagnosis of conversion hysteria was established. In most patients with pseudoseizures, it was necessary for serial evaluations to be performed over a period of months before a definitive diagnosis could be established. Seizures in this group were extremely realistically described, well acted out and frequently of long-standing duration. The difficulty in distinguishing these hysteroid episodes from true convulsions has been pointed out by Liske and Forster. '1 We have observed realistically acted out generalized, focal, adversive, psychomotor, and drop attacks in our patients. Complicating the diagnosis was the fact that these patients were nearly always receiving large amounts of anticonvulsant drugs when first seen, which could fragment the clinical seizure pattern of a true paroxysmal event as well as alter the electroencephalogram. '~ The single most useful diagnostic tool was the persistence of a normal electroencephalogram despite often uncontrolled daily seizure activity. Although not included in this study, patients with true neurogenic seizures who also manifest hysterical conversion are serious management problems, and frequently require extensive psychotherapy. '~ Formal psychologic tests are extremely helpful in screening the pseudoseizure population. We would hesitate, however, to recommend total reliance on psychometric testing when pseudoseizures are suspected, as we do not know the incidence of hysterical features in children with true seizures. The neurologic examination is helpful in distinguishing conversion hysteria from true pathologic dysfunction of the nervous system. Sensory mapping which demonstrates sharply demarcated borders that fail to cross the body's midline, or to follow dermatomes, are often clues to conversion hysteria. Uniocular diplopia almost always indicates hysteria. The red lens cover test may be useful in unmasking false diplopia as well as hysterical uniocular blindness. (Red letters cannot, and black letters can be

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seen through a red lens. When the hysterical blind eye is uncovered and the eye with alleged normal vision is covered with a red glass, only black letters should be visualized.) During the motor examination, an upper motor neuron paretic limb is generally hyperreflexic with long tract signs. The limb immobilized by hysteria usually has normal or absent reflexes and lacks pyramidal signs. An excellent bedside test of a paretic lower limb is the Hoover sign, in which the examiner's palms are placed under the heels of the supine patient. Lifting of the opposite leg results in a downward deflection of the contralateral limb into the palm of the hand. Absence of this volitional effort implies malingering or conversion. Further neurologic features of hysteria are extensively described by DeJong" and in a short monograph by Weintraub. 1:, Prior to this study, we would have expected conversion hysteria to be rare and that, when seen, the patients would nearly always be teen-age girls. This was not borne out by our observations. In the younger population, the ratio between males and females was approximately equal, but by the adolescent years, females did constitute the bulk of patients. In the series reported by Sheffield in 1898,-' one-third were males and two-thirds females: this probably indicates that the ratio has not changed over the past century. Our experience indicates that the neurologic manifestations of childhood hysteria remain common in this century and are still being overlooked by physicians 80 years after L. Emmett Holt, Sr., in his 1898 pediatric text, TM remarked that "hysteria is often apt to be overlooked because its occurrence in children is not considered as often as it should be." REFERENCES

1. Freud S: Some points for a comparative study of organic and hysterical motor paralyses, standard edition, vol I, London+ 1962. 2. SheffieldH: The study of hysteria in childhood, N Y Med J 68:412, 433, 1898. 3. Proctor J: Hysteria in childhood, Am J Ortho psychiatry, 2.8:394, 1958. 4. Keating J+editor: Cyclopedia of the diseases of children, vol 4, Philadelphia, 1891, JB Lippincott Company+ pp 9581007. 5. Rudolph A, editor: Pediatrics, ed 16, New York, 1977, Appleton-Century-Crofts. p 88. 6. PurtellJ, Robins E, and Cohen M: Observations on clinical aspects of hysteria. JAMA 146:902+ 1951. 7. Robins E, and O'Neal P: Clinical features of hysteria in children, with a note on prognosis. Nervous Child 10:246, 1953. 8. StevensH: Conversion hysteria-revisited by the pediatric neurologist, Clin Proc D C Child Hosp 25"27, t969. 9. Lewis M: Child psychiatric consultation in pediatrics, Pediatrics 62:359, 1978.

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10. Jensen R, and Wert A: Conversion hysteria in children, J Lancet 65:172, 1945. 11. LiskeE, and Forster F: Pseudoseizures: A problem in the diagnosis and management of epileptic patients, Neurology 14:41, 1964. 12. Kiloh L, McComas A, and Osselton J: Clinical electroencephalography, London, 1972. Butterworth & Co., Ltd., p 192. 13. WilliamsD, Spiegel H, and Mostofsky D: Neurogenic and

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hysterical seizures in children and adolescents: Differential diagnostic and therapeutic considerations, Am J Psychiatry 135:82, 1978. 14. DeJongR: The neurological examination, ed 3, New York, 1969 Harper & Row, Publishers, pp 989-1015. 15. Weintraub M: Hysteria: A clinical guide to diagnosis, C1BA Clinical Symposia 29, 1977. 16. Holt LE: Diseases of infancy and childhood, New York, 1898, Appleton, p 688.

Brief clinical and laboratory observation Use o f a behavior checkl&t on a pediatric inpatient unit Michael Jellinek, M.D.,* Nina Evans, M.D., and Rona B. Knight, Ph.D., Bronx, N. Y.

EMOTIONAL OR BEHAVIORAL DISORDERS are often hard to define in objective terms and, until recently, have not been given educational priority in some pediatric curricula. On an inpatient service, behavioral and emotional issues often give way to "completing the work-up" and providing acute care. Some children do get referred to a child psychiatrist for developmental evaluation or on the basis of clear indication such as rape, suicide attempt, or bizarre behavior. However, most children over age 3 or 4 years go through the admissions process with little attention to emotional or behavioral issues. In this preliminary study we attempted to develop a set of behavioral questions in a checklist format for 7- to 1 l-year-old inpatients. We hoped that the checklist would facilitate communication between mother and pediatrician, train pediatric houseofficers by providing a short list of questions that fit into the medical review of systems model, and generate a score that would suggest the need for psychiatric consultation. The checklist we studied is a major modification of the Washington Symptom Checklist used by Weinberger and Gregory. l In their study of patients in a child psychiatric outpatient clinic~ they found that the WSCL was useful in From the Department of Pediatrics, Montefiore Hospital and Medical Center. *Reprint address: 15 Rose Garden Circle, Boston, MA 02135

eliciting data from the mother during the clinic intake interview. We modified the WSCL by decreasing the number of questions from 67 to 27, and eliminated open-ended questions in an effort to shorten the time required for administration. Further, we narrowed and changed questions on the basis of our own "review of systems" aproach. For children between the ages of 7 and 11 we "review" five major areas: mood, play, school, friends, and family relations. Our checklist reflects an attempt at integrating clinical concepts and behavioral questions with special concern for ease of administration and brevity. Abbreviations used WSCL: Washington Symptom Checklist SES: socioeconomicstatus A major question in this type of study is whether the mother is an accurate reporter of her child's behavior. Although the mother knows the most about the child, her information is open to major distortion and subjectivity. Gtidewell et al 2 found that mothers were reliable in describing the number, duration, and severity of symptoms. A more recent study :' used a modified WSCL retrospectively to evaluate 30 inpatients who were referred to a child psychiatrist and, as controls, used 60 inpatients who were not referred. The results indicated the potential value of a behavioral checklist; however, virtual-

0022-3476/79/100156 +03500.30/0 9 1979 The C. V. Mosby Co.