LETTERS Limbic Lesions With Recurrent SIR: brain in a Patient Mania mania (mania after lesions) may be the result of injury to limbic or limbic-connected structures in the right hemisphere.’2 We describe the presence of limbic lesions in a patient with a 33-year history of recurrent unipolar mania. Secondary Case Report A 57-year-old left-handed a negative family history woman with of psychiatric for psychiatric care following a one-week history of irritable mood and psychomotor agitation. The patient had a 33-year history of redisorder was admitted current umpolar mania (one manic episode per year, usually during the spring). The manic episodes were characterized by irritability, hyperactivity, self-esteem, loud abusive presspeech, mood-congruent para- inflated sured noid delusions, decreased need overspending, and for sleep. She never had depression, and the manic episodes improved with lithium therapy, although lithium did not prevent the annual recurrence of mania. Clinical and neuro- logical examinations, as well as laboratory studies (endocrine, metabolic, and EEG), were always negative. On admission, the and noncooperative, sured speech, gruent paranoid patient and flight ventricular atrophy, and globus pallidus. giography location confirmed of the vascular of ideas, mood-conand A digital the vermian in the left subtraction presence malformation. anand Discussion We presented a left-handed woman with a 33-year history of recurrent unipolar mania and a left hemisphere vascular malformation. Even though the early age of onset, the long duration of the psychiatric disorder, the pattern of symptom recurrence, and the presence of psychotic symptoms were functional showed disorder, FIGURE a focal suggestive an Mifi of a study lesion involving limbic and limbic-related areas of the left hemisphere (amygdala, internal paffidum, and pulvinar). While it could be argued that manic symptoms and the brain lesion were only related by chance, we have previously reported a high frequency of lesions to limbic or limbic-connected structures in patients with secondary mania.2 Although we was irritable she had pres- delusions, dilation, cerebellar iron deposition 1. have also reported a significant association of secondary mania with right hemisphere lesions,4 the present patient had left hemisphere damage. This reversed cerebral lateralization may be related to atypical brain organization (she was lefthanded). The influence of handedness and side of lesion in the production of secondary mania, however, remains to be systematically examined. To conclude, brain imaging in a subgroup of patients with affective disorders may brain lesions related. that be etiologi- cally might reveal L. BERTHIER, M.D. E. STARKSTEIN, M.D. ROBERT G. ROBINSON, M.D. RAMON LEIGUARDA, M.D. Institute of Neurological Research “Dr. Raul Carrea,” Buenos Aires, MARCELO SERGI0 Argentina; and Psychiatry and Behavioral Department of Sciences, The Johns Hopkins University School Baltimore, MD of Medicine, 12-weighted axial MRI scan showing an arteriovenous malformation involving (left) the left pulvinar (outlined arrow) and (right) the left aniygdala (outlined arrow). Note the iron deposition in the left internal pallidum (left, solid arrow). Inversed 3U insomnia. A psychiatric examination was carried out using a standardized instrument (the modified Present State Exam) and a Mania Rating Scale (MRS).3 The patient met DSM-III-R criteria for a manic congruent episode psychotic with features, MRS score was 19 (definite was prescribed lithium moodand her mania). She (900mg/day) and thioridazine (100 mg/day) and showed improvement. An a gradual MRI scan (figure 1) revealed a left hemisphere arteriovenous malformation involving the amygdala, pallidum, findings and the pulvinar. Additional were a moderate left lateral JOURNAL internal OF NEUROPSYCHIATRY L : ,,q 235 LETTERS that References 1. Cummings JL, Mendez MF: Secondary mania with focal cerebrovascular lesions. AmJ Psychiatry 1984; 141:1084-1087 2. Starkstein SE, Boston JO, Robinson RG: Mechanisms of mania after brain injury: 12 case reports and review of the literature. J Nerv Ment Dis 1988; 176:87-100 3. Bech P, Kastrup M, Rafaelsen OJ: Minicompendium of rating scales for states of anxiety, depression, mania, and schizophrenia, with corresponding DSM-ffl syndromes. Acta Psychiatr Scand 1986; 73(S236):29-31 4. Robinson RG, Boston JO, Starkstein SE, et a!: Comparison of mania and depression after brain injury: causal factors. Am J Psychiatry 1988; 145:172-1 78 digressive. inappropriate. Schizophrenia viscosity, aggression) in LE pa- tients. Yet, a range of conditions and much individual variation occur.3 Within a recent series of LE patients,4 we have found several cases that fulfill DSM-III-R criteria for schizotypal personality disorder. Case Reports Patient 1. in the location, diagnoses his symp- struction). man, At age 24, partial a 35-year-old injury at age last two years, have been used toms complex often and various to describe (e.g., atypical psychosis, paranoid schizophrenia, fied personality disorder). and major tient has complied with anticonvulsant medication irregularly, due to his belief acute and unspeci- a 20-year-old was delivered as the second twin a difficult 18-hour labor. Complex absence, and major motor sei6. EEGs revealed bitemporal lobe foci. at age zures began lateral anterior the last few years, the patient has to develop alternative identities. He uses these identities to confuse people he believes are prejugone to great diced against lengths him. are conspiring him by making He fears to discredit that others identities, vague, and he becomes tangential. ses have been used toms (e.g., paranoid angry, Several to intermittent explosive disorder, ganic personality disorder). between these One way to determine anistic elements would pare his symp- and or- be to com- challenges (e.g., amphetamines). neuroleptics These drugs been shown to improve be disorders.3 shared mech- to pharmacological and of biological degree and have of correlates of (e.g., seizure focus of brain tissue de- Longitudinal studies of selected DSM-III-R subgroups may yield further insights. For example, in LE patients with schizotypal DSM-III-R personality disorder, is there a progression of symptoms with time toward frank psychosis? Such a progression would lend further credence to the notion that certain SSDs are transitional forms, and a longitudinal study would provide a valuable opportunity for prospective study psychosis of the mechanisms of development. 1. Siever U, Gunderson schizotypal personality: and current status. JG: The search for a historical origins Compr Psychiatry SC, Geschwind N: The interictal syndrome of temporal lobe epi- lepsy. Arch Gen Psychiatry 1975; 32:15801586 3. Stevens JR: Psychiatric aspects of epilepsy. J Clin Psychiatry 1988; 49:49-57 4. Csernansky may responses studies 2. Waxman behavior Discussion Approaches that are used in the study of SSDs and LE have much to offer each other. The etiologies and pathogenic mechanisms of schizophrenia and SSDs are unknown. For LE, the etiology is often known, and mechanisms to explain symptom production may be more readily investigated. Certain anatomical or mechanisms symptoms 1983; 24:199-212 digressive, diagno- to describe disorder, the References and insult his problems known everyone. He has no social contacts. When one inquires about his multiple common 20. motor seizures began. EEGs revealed left temporal lobe and bilateral anterior temporal lobe foci. Since age 32, the pa- 236 of psychiatrists neurochemical The patient, suffered a head and audi- respectively, SSD patients.5 For LE research, the subgrouping of patients using DSM-HI-R personality disorder criteria would aid attention Over SIR: Schizophrenia spectrum disorders (SSDs) were first noted among the family members of schizophrenics. Now, many of the DSM-III-R cluster A personality disorders are considered to be within this “spectrum.” However, before SSDs are considered unique syndromes that occur only in relation to functional psychiatric disorders, SSD researchers should consider interictal psychopathology seen in some patients with limbic epilepsy (LE). Waxman and Geschwind2 emphasized certain characteristic symptoms (e.g., bate, psychopathology partial, Limbic by fre- and affect is exaggerated He admits to vague His Patient 2. The patient, Spectrum and his seizures tory hallucinations and intense sexual fantasies. He has few acquaintances and no friends. The patient has come to the man, after Disorder Epilepsy he can control quent masturbation. The patient is circumstantial JG, Leiderman DB, Mandabach M, eta!: Psychopathology and limbic epilepsy: relationships to seizure variables and neuropsychological function. Epilepsia (in press) 5. Schulz SC, Cornelius J, Jarret DB: Pharmacodynamic probes in personality disorders. Psychopharmacol Bull 1987; 23:337-351 Jorn. G. CSERNANSKY, DEBORAH M.D. B. LEIDERMAN, M.D. JEFF GOLDMAN Palo Alto V.A. Medical Center, Palo Alto, California; Department of Psychiatry and Behavioral Sciences, Stanford School of Medicine, California; National Bethesda, University Stanford, and Epilepsy Section, Institutes of Health, Maryland exacer- VOLUME 2 #{149} NUMBER 2 #{149} SPRING 1990