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"Rheumatic" Schizophrenia: Title and subTitle BreakAn Epidemiological Study

NANCY M. WERTHEIMER, Ph.D.
[+] Author Affiliations

Submitted for publication Nov. 21, 1960.


Arch Gen Psychiatry. 1961;4(6):579-596. doi:10.1001/archpsyc.1961.01710120049006
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It seems clear that rheumatic fever can involve the nervous system. Chorea is the most commonly recognized form of such involvement, but neurological symptoms suggestive of meningitis and encephalitis3 as well as psychological symptoms13,37 have also been observed. For nearly a century the literature has recognized the occasional presence of psychosis as an apparent symptom of rheumatic fever,31 and from time to time the similarity of some of these psychoses to schizophrenia has been remarked.4,19,23,27,31

The evidence of the present study suggests that far more may be involved than an occasional resemblance between rheumatic brain symptoms and schizophrenia. The hypothesis tested and supported here is that a large portion of schizophrenics, including many who present a typical schizophrenic course and symptomatology with no outstanding organic symptoms and no traditional signs of rheumatic fever, may nonetheless actually have psychoses

REFERENCES

The age at initial rheumatic involvement is considered especially important in determining which symptom pattern will appear, for as Roth26 indicates, the state of a tissue at the time of initial rheumatic involvement may determine whether or not it will be affected, but, once affected, the tissue may for some time remain in a state which disposes it to further rheumatic attack. It is possible 4,8 that rheumatic attacks are not truly discrete events, but simply exacerbations of a continuous, underlying process (or tissue sensitivity). Which tissues will be involved may be almost completely determined at the time of the initial "sensitization," but the appearance of clinical manifestations may vary widely for different tissues.
Age-specificity can be used to explain cases of "pure" Sydenham's chorea. These would hypothetically be cases where rheumatic disease attacked a child at a time when his particular developmental pattern was "ripe" for the appearance of chorea as a symptom, but already "overripe" for the (typically earlier-occurring) symptom of rheumatic heart disease. This hypothesis permits "pure" chorea to be seen as a true rheumatic symptom, yet anticipates the empirical finding that such cases do not seem to develop subsequent rheumatic heart disease.16 The fact that both chorea and heart symptoms do frequently occur in the same rheumatic child presumably indicates that many hosts go through an age when they are susceptible to both symptoms.
While the grimacing of schizophrenia may be somewhat different from that observed in typical chorea, it is assumed here that a similar brain process could underlie both, and that this process might in each case be a sign of rheumatic brain involvement. In his discussion of the "spasmodic phenomena" in dementia praecox, Kraepelin18 (p. 83) mentions that the grimacing and twitching observed "remind one of the corresponding disorders of choreic patients."
There is a sex difference (not shown in Table 1) which suggests that schizophrenic grimacing may be related to the occurrence of a rheumatic attack during puberty, rather than to its occurrence at a particular age, as such. Schizophrenic grimacing was most common (46% of 13 cases) in the female patients whose first rheumatic attack was noted between 9 and 16. Rheumatic onset at other ages rarely "produced" grimacing (12% of 26 cases). For males, schizophrenic grimacing was most commonly noted when the first rheumatic attack occurred a little later, between ages 13 and 18 (41% of 17 cases). Only 24% of the remaining 59 males with overt rheumatic history showed grimacing. This sex difference runs roughly parallel to the sex difference in age at puberty.
Definition A appears to tap largely those "rheumatic" schizophrenics whose rheumatic lesions were initiated in childhood (Section II,B). Definition C presumably taps cases with later rheumatic onset (Section I). Thus the irreversible lesions (poor prognosis) seem largely associated with later rheumatic onset—a finding also consistent with the very different outcomes seen in chorea and in grimacing schizophrenia (Section III,F).
Chapman,5 Gerstley,9 Leys,19 and Shaskan 27 all present data at least somewhat suggestive of excess representation of first siblings in chorea, although their data were not gathered or analyzed primarily with this in mind.
See p. 589.
Aycock, W. L., and Eaton, P.:  Epidemiology of Infantile Paralysis: Relation Between Multiple Cases in the Same Family , Amer. J. Hyg. 5:724-732, 1925;.
Benedict, P. K.:  Socio-Cultural Factors in Schizophrenia , in Schizophrenia: A Review of the Syndrome , edited by L. Bellak and P. K. Benedict, New York, Logos Press, 1958;.
Brainerd, H. D., and Sokolow, M.:  The Differential Diagnosis of Rheumatic Fever and Infections of the Central Nervous System , J. Pediat. 34:204-212, 1949;.
Breutsch, W. L.:  Chronic Rheumatic Brain Disease as a Possible Factor in the Causation of Some Cases of Dementia Praecox , Amer. J. Psychiat. 97:276-296, 1940;.
Chapman, A. H., et al.:  A Psychosomatic Study of 8 Children with Sydenham's Chorea , Pediatrics 21:582-595, 1958;.
Coburn, A. F., and Moore, L. V.:  The Independence of Chorea and Rheumatic Activity , Amer. J. Med. Sci. 193:1-3, 1937;.
Collins, S. D.:  The Incidence of Rheumatic Fever as Recorded in General Morbidity Surveys of Families , Public Health Rep. (Supp. 198) , 1947;.
Feinstein, A. R., and Spagnuolo, M.:  Mimetic Features of Rheumatic Fever Recurrences , New Engl. J. Med. 262:533-540, 1960;.
Gerstley, J. R., et al.:  Chorea: Is It a Manifestation of Rheumatic Fever? J. Pediat. 6:42-50, 1935;.
Grinker, R. R., and Bucy, P. C.: Neurology, Springfield , Ill., Charles C Thomas, Publisher, 1949;
Howie, D. L.:  Some Pathological Findings in Schizophrenics , Amer. J. Psychiat. 117:59-63, 1960;.
Huston, P. E., and Pepernick, M. C.:  Prognosis in Schizophrenia, in Schizophrenia : A Review of the Syndrome , edited by L. Bellak and P. K. Benedict, New York, Logos Press, 1958;.
Haskell, R. H.:  Mental Disturbances Associated with Acute Articular Rheumatism , Amer. J. Insanity 71:361-381, 1914;.
Hollingshead, A. B., and Redlich, F. C.: Social Class and Mental Illness , New York, John Wiley & Sons, Inc., 1958;.
Jacobsson, E.:  Rheumatic Fever with Chorea Minor , Acta Paediat. 33: ( (Supp. 7) ), 1946;.
Jones, T. D., and Bland, E. F.:  Clinical Significance of Chorea as a Manifestation of Rheumatic Fever , J.A.M.A. 105:571-577, 1935;.
Kagan, B. M., and Mirman, B.:  Sydenham's Chorea, a Syndrome for Differential Diagnosis , J. Pediat. 31:322-332, 1947;.
Kraepelin, E.: Dementia Praecox and Paraphrenia , Edinburgh, E. & S. Livingstone, Ltd., 1919;.
Leys, D.:  Rheumatic Encephalopathy, Edinburgh Med. J. 53:444-449, 1946;.
Mettler, F. A.:  Perceptual Capacity, Functions of the Corpus Striatum, and Schizophrenia , Psychiat. Quart. 29:89-111, 1955;.
Morbidity and Mortality, Annual Supp. for 1952, 1953, and 1955, U.S. National Office of Vital Statistics.
Patients in Mental Institutions, 1952, 1953, and 1955, U.S. National Institute of Mental Health.
Neubuerger, K. T.:  The Brain in Rheumatic Fever , Dis. Nerv. Syst. 8:1-4, 1947;.
Paul, J. R.: The Epidemiology of Rheumatic Fever , New York, American Heart Association, Inc., 1957;.
Rantz, L. A., et al.:  Hemolytic Streptococcal Infection in Childhood , Pediatrics 12:498-515, 1953;.
Roth, R., et al.:  Heart Disease in Children: (A) Rheumatic Group; (I) Certain Aspects of the Age at Onset and of Recurrences in 488 Cases of Juvenile Rheumatism Ushered in by Major Clinical Manifestations , Amer. Heart J. 13:36-60, 1937;.
Shaskan, D.:  Mental Changes in Chorea Minor , Amer. J. Psychiat. 95:193-202, 1938;.
Taranta, A.:  Relation of Isolated Recurrences of Sydenham's Chorea to Preceding Streptococcal Infections , New Engl. J. Med. 260:1204-1209, 1959;.
Taranta, A., and Stollerman, G. H.:  Relationship of Sydenham's Chorea to Infection with Group A Streptococci , Amer. J. Med. 20:170-175, 1956;.
Rheumatic Fever: A Symposium, edited by L. Thomas, Minneapolis, Univ. of Minnesota, 1952.
Van der Horst, L.:  Rheumatism and Psychosis, Digest Neurol. Psychiat. 15:399-416, 1947;.
Vogt, C., and Vogt, O.:  Precipitating and Modifying Agents in Chorea , J. Nerv. Ment. Dis. 116:601-607, 1952;.
Weiner, H.:  Diagnosis and Symptomatology, in Schizophrenia : A Review of the Syndrome , edited by L. Bellak and P. K. Benedict, New York, Logos Press, 1958;.
Wertheimer, N.:  The Differential Incidence of Rheumatic Fever in the Histories of Paranoid and Nonparanoid Schizophrenics , J. Nerv. Ment. Dis. 125:637-641, 1957;.
Wertheimer, N.: A Psychiatric Follow-Up of Rheumatic and Choreic Children and Their Siblings, to be published.
Wilson, M. G.: Rheumatic Fever , New York, Commonwealth Fund, 1940;.
Wilson, S., and Bruce, A.: Neurology, Baltimore , The Williams & Wilkins Company, 1955;.

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Country-Specific Mortality and Growth Failure in Infancy and Yound Children and Association With Material Stature

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The age at initial rheumatic involvement is considered especially important in determining which symptom pattern will appear, for as Roth26 indicates, the state of a tissue at the time of initial rheumatic involvement may determine whether or not it will be affected, but, once affected, the tissue may for some time remain in a state which disposes it to further rheumatic attack. It is possible 4,8 that rheumatic attacks are not truly discrete events, but simply exacerbations of a continuous, underlying process (or tissue sensitivity). Which tissues will be involved may be almost completely determined at the time of the initial "sensitization," but the appearance of clinical manifestations may vary widely for different tissues.
Age-specificity can be used to explain cases of "pure" Sydenham's chorea. These would hypothetically be cases where rheumatic disease attacked a child at a time when his particular developmental pattern was "ripe" for the appearance of chorea as a symptom, but already "overripe" for the (typically earlier-occurring) symptom of rheumatic heart disease. This hypothesis permits "pure" chorea to be seen as a true rheumatic symptom, yet anticipates the empirical finding that such cases do not seem to develop subsequent rheumatic heart disease.16 The fact that both chorea and heart symptoms do frequently occur in the same rheumatic child presumably indicates that many hosts go through an age when they are susceptible to both symptoms.
While the grimacing of schizophrenia may be somewhat different from that observed in typical chorea, it is assumed here that a similar brain process could underlie both, and that this process might in each case be a sign of rheumatic brain involvement. In his discussion of the "spasmodic phenomena" in dementia praecox, Kraepelin18 (p. 83) mentions that the grimacing and twitching observed "remind one of the corresponding disorders of choreic patients."
There is a sex difference (not shown in Table 1) which suggests that schizophrenic grimacing may be related to the occurrence of a rheumatic attack during puberty, rather than to its occurrence at a particular age, as such. Schizophrenic grimacing was most common (46% of 13 cases) in the female patients whose first rheumatic attack was noted between 9 and 16. Rheumatic onset at other ages rarely "produced" grimacing (12% of 26 cases). For males, schizophrenic grimacing was most commonly noted when the first rheumatic attack occurred a little later, between ages 13 and 18 (41% of 17 cases). Only 24% of the remaining 59 males with overt rheumatic history showed grimacing. This sex difference runs roughly parallel to the sex difference in age at puberty.
Definition A appears to tap largely those "rheumatic" schizophrenics whose rheumatic lesions were initiated in childhood (Section II,B). Definition C presumably taps cases with later rheumatic onset (Section I). Thus the irreversible lesions (poor prognosis) seem largely associated with later rheumatic onset—a finding also consistent with the very different outcomes seen in chorea and in grimacing schizophrenia (Section III,F).
Chapman,5 Gerstley,9 Leys,19 and Shaskan 27 all present data at least somewhat suggestive of excess representation of first siblings in chorea, although their data were not gathered or analyzed primarily with this in mind.
See p. 589.
Aycock, W. L., and Eaton, P.:  Epidemiology of Infantile Paralysis: Relation Between Multiple Cases in the Same Family , Amer. J. Hyg. 5:724-732, 1925;.
Benedict, P. K.:  Socio-Cultural Factors in Schizophrenia , in Schizophrenia: A Review of the Syndrome , edited by L. Bellak and P. K. Benedict, New York, Logos Press, 1958;.
Brainerd, H. D., and Sokolow, M.:  The Differential Diagnosis of Rheumatic Fever and Infections of the Central Nervous System , J. Pediat. 34:204-212, 1949;.
Breutsch, W. L.:  Chronic Rheumatic Brain Disease as a Possible Factor in the Causation of Some Cases of Dementia Praecox , Amer. J. Psychiat. 97:276-296, 1940;.
Chapman, A. H., et al.:  A Psychosomatic Study of 8 Children with Sydenham's Chorea , Pediatrics 21:582-595, 1958;.
Coburn, A. F., and Moore, L. V.:  The Independence of Chorea and Rheumatic Activity , Amer. J. Med. Sci. 193:1-3, 1937;.
Collins, S. D.:  The Incidence of Rheumatic Fever as Recorded in General Morbidity Surveys of Families , Public Health Rep. (Supp. 198) , 1947;.
Feinstein, A. R., and Spagnuolo, M.:  Mimetic Features of Rheumatic Fever Recurrences , New Engl. J. Med. 262:533-540, 1960;.
Gerstley, J. R., et al.:  Chorea: Is It a Manifestation of Rheumatic Fever? J. Pediat. 6:42-50, 1935;.
Grinker, R. R., and Bucy, P. C.: Neurology, Springfield , Ill., Charles C Thomas, Publisher, 1949;
Howie, D. L.:  Some Pathological Findings in Schizophrenics , Amer. J. Psychiat. 117:59-63, 1960;.
Huston, P. E., and Pepernick, M. C.:  Prognosis in Schizophrenia, in Schizophrenia : A Review of the Syndrome , edited by L. Bellak and P. K. Benedict, New York, Logos Press, 1958;.
Haskell, R. H.:  Mental Disturbances Associated with Acute Articular Rheumatism , Amer. J. Insanity 71:361-381, 1914;.
Hollingshead, A. B., and Redlich, F. C.: Social Class and Mental Illness , New York, John Wiley & Sons, Inc., 1958;.
Jacobsson, E.:  Rheumatic Fever with Chorea Minor , Acta Paediat. 33: ( (Supp. 7) ), 1946;.
Jones, T. D., and Bland, E. F.:  Clinical Significance of Chorea as a Manifestation of Rheumatic Fever , J.A.M.A. 105:571-577, 1935;.
Kagan, B. M., and Mirman, B.:  Sydenham's Chorea, a Syndrome for Differential Diagnosis , J. Pediat. 31:322-332, 1947;.
Kraepelin, E.: Dementia Praecox and Paraphrenia , Edinburgh, E. & S. Livingstone, Ltd., 1919;.
Leys, D.:  Rheumatic Encephalopathy, Edinburgh Med. J. 53:444-449, 1946;.
Mettler, F. A.:  Perceptual Capacity, Functions of the Corpus Striatum, and Schizophrenia , Psychiat. Quart. 29:89-111, 1955;.
Morbidity and Mortality, Annual Supp. for 1952, 1953, and 1955, U.S. National Office of Vital Statistics.
Patients in Mental Institutions, 1952, 1953, and 1955, U.S. National Institute of Mental Health.
Neubuerger, K. T.:  The Brain in Rheumatic Fever , Dis. Nerv. Syst. 8:1-4, 1947;.
Paul, J. R.: The Epidemiology of Rheumatic Fever , New York, American Heart Association, Inc., 1957;.
Rantz, L. A., et al.:  Hemolytic Streptococcal Infection in Childhood , Pediatrics 12:498-515, 1953;.
Roth, R., et al.:  Heart Disease in Children: (A) Rheumatic Group; (I) Certain Aspects of the Age at Onset and of Recurrences in 488 Cases of Juvenile Rheumatism Ushered in by Major Clinical Manifestations , Amer. Heart J. 13:36-60, 1937;.
Shaskan, D.:  Mental Changes in Chorea Minor , Amer. J. Psychiat. 95:193-202, 1938;.
Taranta, A.:  Relation of Isolated Recurrences of Sydenham's Chorea to Preceding Streptococcal Infections , New Engl. J. Med. 260:1204-1209, 1959;.
Taranta, A., and Stollerman, G. H.:  Relationship of Sydenham's Chorea to Infection with Group A Streptococci , Amer. J. Med. 20:170-175, 1956;.
Rheumatic Fever: A Symposium, edited by L. Thomas, Minneapolis, Univ. of Minnesota, 1952.
Van der Horst, L.:  Rheumatism and Psychosis, Digest Neurol. Psychiat. 15:399-416, 1947;.
Vogt, C., and Vogt, O.:  Precipitating and Modifying Agents in Chorea , J. Nerv. Ment. Dis. 116:601-607, 1952;.
Weiner, H.:  Diagnosis and Symptomatology, in Schizophrenia : A Review of the Syndrome , edited by L. Bellak and P. K. Benedict, New York, Logos Press, 1958;.
Wertheimer, N.:  The Differential Incidence of Rheumatic Fever in the Histories of Paranoid and Nonparanoid Schizophrenics , J. Nerv. Ment. Dis. 125:637-641, 1957;.
Wertheimer, N.: A Psychiatric Follow-Up of Rheumatic and Choreic Children and Their Siblings, to be published.
Wilson, M. G.: Rheumatic Fever , New York, Commonwealth Fund, 1940;.
Wilson, S., and Bruce, A.: Neurology, Baltimore , The Williams & Wilkins Company, 1955;.

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