Pseudocyesis in Organic Mood Disorders Six Cases
STEPHEN F. SIGNER, M.D., ROBERT
P.
WEINSTEIN, M.D.
A. MUNOZ, M.D., J. FERNANDO BAYARDO, M.D. MARK R. KATZ, M.D., LAURENCE R. SABEN, M.D.
RODRIGO
Pseudocyesis. the delusion of pre~nancy. has had an uncertain nosolo~y. primarily because of the concentration on the content of the beliefs and lack of interest in the underlyin~ phenomenolo~y. Six patients with a major mood disorder caused by cerebral dysfunction are presented in this article. The delusion is reviewed with respect to the entities it overlaps. and the clinical manifestations are related to the mood disorders. Although no clear neuroanatomic localization was possible with this ~roup of patients. there may be some association with desomatization caused by parietal lobe dysfunction.
A
lthough a large number of case reports from the past have been accumulated, pseudocyesis has become uncommon in industrialized societies with the exception of a large series from West and South Africa. Because of the importance of determination of succession in many cultures, few delusions have shown such a clear influence on the course of history (Mary Tudor, Natalie of Serbia). The modem term, coined by Good in 1823 from the Greek root words for "false pregnancy," has replaced the variants in the older literature. I Pseudocyesis has been defined as a state in which a Received June 3. 1991; revised August 30. 1991; accepted September 6. 1991. From the Depanment of Psychially, University of California. San Diego. Address reprint requests to Dr. Signer, Psychiatric Centers at San Diego, 15725 Pomerado Road. Suite 206, Poway (San Diego), CA 92064. Copyright © 1992 The Academy of Psychosomatic Medicine.
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woman believes herself to be pregnant, developing the commonly occurring symptoms and signs (i.e., amenorrhea, gradual abdominal enlargement, breast changes, fetal movement, morning nausea and vomiting, and weight gain) in the absence of actual pregnancy. Although defined as a false belief (delusion) in that the patient insists on the reality of the pregnancy despite evidence to the contrary, pseudocyesis has been considered a conversion symptom that is exclusive of pseudopregnancy (medical illness), simulated pregnancy (malingering), or hallucinatory pregnancy (psychosis).2 As such, pseudocyesis is regarded as a paradigm of psychosomatic disorders even though the phenomenology has been quite variably and poorly recorded. The traditional emphasis on presumed psychodynamic content and conflicts ignores and downplays the more modem interest in the importance of phenomenologic features in determining the nosology. At this point, it is PSYCHOSOMATICS
Signer et al.
inappropriate to allow theoretical considerations to interfere with the explorations of the boundaries of a distinct syndrome. This article presents six new cases of the delusion of pregnancy caused by brain injury in the context of a mood disorder (organic mood disorder) and reviews the other disorders in which this false belief can occur. Case Reports Case 1. Ms. A.• a 39-year-old. right-handed separated woman of mixed North American Indian and French-Canadian descent. had three children (22. 16. and 7 years of age). She presented to her general doctor with 4 months of amenorrhea. increasing abdominal pain. and the sensation of internal movement. and he confirmed her belief that she was pregnant. After a negative pregnancy test. a pelvic ultrasound was done that showed a normal nongravid abdomen. When Ms. A. was told she was not pregnant, she became grossly agitated. believing there was some sort of monster or worms inside her. On admission. she complained of numbness all over her body, depressed mood. sleep disturbance. and hallucinations commanding her to harm herself. Ms. A.·s mother had recurrent depression and other family members had alcoholism. Ms. A. had had a brutal childhood with physical and sexual abuse. She became pregnant after a rape at the age of 17 years. and the child was raised by her mother; a second child. born when she was 23 years old. was given to the care of the Children's Aid Society (CAS). She separated from her alcoholic husband 3 years after the birth of her last child and found the repeated contact with him very stressful. One month before the onset of symptoms. the supervision of a CAS social worker, whom she liked, ended. On examination there was no galactorrhea and her uterus was normal. Urine pregnancy. betahuman chorionic gonadotropin (HCG). and dexamethasone suppression tests were negative. One measure of serum prolactin was elevated at 36 ~g/L. Thyroid function tests were normal. The brain cr scan was normal; the EEG showed bilateral mild slow wave disturbances. On the Wechsler Adult Intelligence Scale-Revised (WAIS-R), the verbal subscale was 53. the performance subscale was 72. and the full scale was 58; the Wechsler Memory Quotient was 52. Her educational level was below VOLUME 33· NUMBER 3· SUMMER 1992
that of the 3rd grade in elementary school. She was treated with fluphenazine decanoate 50 mg im twice a month for a diagnosis of delusional disorder. Ms. A. was readmitted 6 weeks later with depressed mood; diurnal variation; crying; agitation; anorexia; initial. middle. and terminal insomnia; fatigue; loss of interest; social withdrawal; poor concentration; and homicidal and suicidal ideation. She complained again of the feelings of numbness and milky discharge from her right breast. although none could be expressed. but did not have the delusion of false pregnancy. At that point the diagnosis of unipolar depression was much clearer. and she responded to treatment with imipramine 125 mg qd and haloperidol 10 mg qd. Case 2. Ms. B.• a 29-year-old. right-handed twicemarried Hispanic woman, claimed to have had 30 children. mostly twins, after eight pregnancies. She believed that she was again pregnant with twins and was experiencing amenorrhea. abdominal protrusion. heartburn and nausea, weight gain. breast tenderness. and the sensation of movement; there were two episodes of false labor. Ms. B. achieved menarche at age 12 years with irregular cycles. having mild postpartum dysphoria. She said all the children were taken from her at birth. Her medical history was noncontributory aside from past binge drinking and drug abuse, including cannabis. narcotics, hallucinogens. psychostimulants, sedatives. and solvents. The patient alternated between periods of depression and elation. The depressions lasted 1-2 weeks with decreased appetite and weight. decreased energy, increased sleep. and psychomotor slowing. Hypomanic states were longer lasting. with increased activity, decreased sleep. and grandiosity. She had been hyperactive and destructive as a child; there were several hospitalizations for mania with grandiose delusions. Ms. B. was elated, with rapid. pressured speech and thought form marked by tangentiality. circumstantiality. and derailment. She had some unorganized ideas of surveillance. but denied erotic. grandiose, control, substitution. or other somatic delusions. The digit span was six forward but none in reverse; the reversal of other learned sequences was slowed. Her language function was normal. although verbal fluency was decreased; confabulation appeared in a test of short-term memory. The ReyOsterreith figure showed distortion on the right. segmental approach. with perseveration and fusion with previous form. A clock drawing showed perseveration to the number 19; the Luria hand se317
Pseudocyesis in Organic Mood Disorders
quence demonstrated verbal-motion dissociation on the right. The motor examination showed rightsided drift. increased tone. and retlexes. The pregnancy test was negative. Treatment with lithium. tluphenazine. and amitriptyline stabilized her mood state. and the delusion disappeared. Case 3. Ms. c.. a 68-year-old. right-handed former clerk. claimed six previous marriages and seven children. She threatened others with knives and was preoccupied with cutting up objects. The patient was elated. with an increased level of activity. pressured rapid speech. derailment. tangentiality. and circumstantiality. Ms. C. denied having been depressed although some mixed affective symptoms were present; she characterized herself as a "super energy" person. Ms. C. claimed that a younger fundamentalist preacher had married her in May (erotomania) and that she was 2 months pregnant. She claimed morning sickness. increased weight. and breast fullness; she soon expected to feel movement and wanted a caesarean section. Menarche had occurred at age 13 years and she denied any menopausal symptoms. Ms. C.·s father was an "inventor" and alcoholic. as were many members of his family. She had diabetes mellitus. hypenension. and coronary anery disease; her medications included chlorpropamide. c1onidine. diltiazem. and nitroglycerin. to which lithium was added with control of the symptoms. Her attention was mildly impaired; language was normal although word list generation was markedly decreased; shon-term memory was impaired with confabulation; and Taylor figure showed a segmental strategy. with recall demonstrating a loss of form. The neurologic examination showed extinction of double simultaneous stimulation in the left visual field. decreased upward gaze. decreased saccades to the left. left facial weakness. and increased tone and grasp retlex on the left with distraction maneuvers. The CT scan demonstrated an infarction in the right basal ganglia and anterior limb of the internal capsule and frontal lobe. The mood cycles diminished with the use of lithium. and the delusions were controlled with thioridazine. Case 4. Ms. D.• a 27-year-old. right-handed woman. had prolonged intraoperative anoxia with subsequent coma at age 17 years. She had noted some right-handed weakness and difficulty in writing. Ms. D. had been hospitalized several times. the last for agitation and persecutory delusions. with auditory hallucinations and thought broadcasting. In 3111
addition to some periods of depression. the patient had episodes of elation. with promiscuity and impulsive overspending. The patient believed herself to be pregnant without intercourse. claiming amenorrhea. breast fullness and enlargement. increased appetite with a desire for healthful food. and firmness across her abdomen. Menarche had been achieved at age 12 years. premenstrual affective changes were denied. and she claimed previous pregnancy without intercourse lasting 10 months and delivering a "goldencolored" boy. The pregnancy test was negative. and the family history was noncontributory. She had abused cocaine during two extended periods. Two seizures of unknown phenomenology had occurred. She was treated with lithium. carbamazepine. and thiothixene. with poor response either of the elated mood or symptoms of pseudocyesis. Her attention was normal although she performed both parts of the Trail-Making Test below the 50th percentile; language was normal and there was a marked disparity between the word list generation for concrete objects (animals 17) and letters (using F. A. and S. total = 25). Confabulation appeared on testing shortterm memory. which improved with repetition and cuing; writing showed micrographia of the frontal lobe variety; perseveration was present in drawings; and the Rey-Osterreith figure showed loss of detail and perseveration on recall. The neurologic examination demonstrated difficulty moving the eyes past the midline to the left. decreased left optokinetic nystagmus; decreased strength on the left. increased tone on the left with distraction maneuvers. and increased reflexes on the left. Case 5. Ms. E.. a 43-year-old. right-handed housewife. was admitted in an agitated psychotic state after several weeks of deterioration. She had been hospitalized in similar circumstances in the past. She had had episodes of depression. withdrawal. and poor self-care as well as others of increased activity. frequently harassing calls or letters to others. and increased spending. In addition to ideas that others could control her mind and were accusing her of stealing. Ms. E. believed that she was pregnant because of amenhorrhea. The patient thought her appetite decreased with occasional vomiting. increased weight. abdominal firmness. increased breast size with galactorrhea. and fetal movements. The patient was born I month premature. delivered by high forceps. and at 6 months had a viral infection that caused paralysis of the palate. tlatPSYCHOSOMATICS
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tened affect, slow development of developmental milestones, and learning and speech problems. She had diabetes mellitus, asthma, and hypothyroidism. Ms. E. did not have children, although she had had several miscarriages and a "missed" abortion led to one hospitalization. She had a loaded pedigree for affective disorders. The patient showed a psychomotor retarded parkinsonian appearance. Attention and memory were impaired with considerable perseveration. Right-sided neglect was present on the Taylor figure. Ms. E. demonstrated an increased left palpebral fissure, dysarthria, left-sided clumsiness, increased left-sided reflexes, a small left hand, bilateral grasp reflex, and a positive glabellar tap. Psychological testing showed full-scale IQ to be 69 (verbal 62, performance 67). The thyroid-stimulating hormone was 13.9. The EEG showed diffuse slowing and the CT scan prominent cerebral atrophy. The delusions disappeared with the use of neuroleptics, which had to be stopped due to akathisia and secondary parkinsonian symptoms. Case 6. Ms. E, a 59-year-old, right-handed woman with four children, was admitted with a psychosis characterized by depression, loss of appetite, insomnia, lethargy, suicidal ideation, auditory hallucinations. and persecutory delusions. Over the past 30 years she had had over to psychiatric hospitalizations for both depressive and manic episodes. requiring treatment with electroconvulsive therapy (ECT). neuroleptics, lithium, and carbamazepine at various times. Ms. E had been prescribed lithium and fluphenazine. pilocarpine for glaucoma. digoxin for cardiac arrhythmia, and theophylline for chronic obstructive lung disease. In addition to hypertension. the patient had small-cell carcinoma of the lung treated with radiotherapy and chemotherapy. Ms. E had the recent onset of the belief that she was 9 months pregnant with weight gain of 10 Ibs., abdominal swelling, and the sensation of fetal movements and false labor; she was unsure whether to attribute amenorrhea of 8 years' duration to the "pregnancy" or to "change of life." The patient was irritable and uncooperative. Her affect was somewhat blunted and mood depressed, and hallucinations were not endorsed. The patient was inattentive with psychomotor slowing, and mild deficits in language and memory were present; speech was aprosodic. The neurologic examination did not demonstrate lateralizing signs. Her full-scale WAIS-R was 83. The CTshowed multiple small areas of infarction in the brain stem VOLUME 33· NUMBER 3· SUMMER 1992
and frontal lobes. The pregnancy test was negative; follicle-stimulating hormone (FSH) was 8 mIU/ml, luteinizing hormone (LH) was I mIU/ml, and prolactin was 44 mIU/ml. The mood changes and psychosis remitted with lithium, haloperidol, and clonazepam.
DISCUSSION All of the patients exhibited the delusion of pregnancy with the appropriate physical symptoms in the setting of a psychosis associated with a major mood disorder due to identifiable organic deficits. All had abdominal symptoms including the sensation of fetal movement. Ms. A., who evolved more neurovegetative symptoms of depression over time, was moderately mentally retarded with diffuse EEG changes. Ms. B. exhibited a course of alternating depression and elation with the presence of delusion in the latter phase; evidence of left-hemispheric injury was apparent on the cognitive and neurologic examinations. Ms. C. had a right basal ganglia infarction. Ms. D. had an anoxic encephalopathy with predominantly right hemispheric signs. Ms. E. had several causes for brain injury in the first years of life; there were predominantly right-hemispheric signs present. Ms. F., while ill with schizoaffective disorder, had the new onset of the delusion with a dementia due to white matter infarction from embolic disease. In this group with antecedent organic features, the association seems to be with a severe mood disorder with psychosis, rather than with any lateralized (three right-. one lefthemisphere; two diffuse injury) neuropsychological features, although distortion of body image may be more common with right-hemispheric injury. Aside from Ms. A., who showed a major depression with psychosis, all the cases demonstrated bipolar mood disorders with psychosis. Demographic Characteristics There are no clear demographic or sociocultural indicators for the population at risk. Subjects have ranged in age from 5 to 79 years, 319
Pseudocyesis in Organic Mood Disorders
with an average of 33 years; 73% were between 15 and 39 years; and 17% were postmenopausal. Most of the patients were married, 37% had been pregnant previously, and 5% had recurrent pseudocyesis. The symptoms had lasted 9 months in 43%,1.3.4 rarely persisting for 1-2 years. Clinical Manifestations The most common symptoms and signs include the following (from a combined series):1.3 menstrual abnormalities (oligomenorrhea, and uncommonly amenorrhea; 69%), gradual abdominal enlargement (68%), and the sensation offetal movement (63%). A history of infertility (59%), breast changes (enlargement, tenderness, pigmentation; 59%), weight gain (44%), galactorrhea (42%), and cervical changes (40%) have also been noted.u.4 Morning sickness may appear before the first missed menstrual period and the quickening occurs earlier than in a real pregnancy. The abdomen is usually rounded with uniform distension and tense, rubbery tone and weight gain is usually greater than normal. The umbilicus is inverted, the cervix firm, the uterus not enlarged, and no fetal parts are palpable. The abdominal distention, due probably to changes in tone of the diaphragm and abdominal muscles, lordosis, and possibly omental and abdominal fat, and bowel distention, usually disappears with steady pressure, Bargen's maneuver, anesthesia, or muscle relaxants. 4 .5 False labor occurred in I% of a group of modem series' and 30% of the historical group. 4 Fetal movements, atypical for their intensity, period of onset, or pattern, may be so violent as to be socially disruptive and presumably are due to abdominal muscle contractions. I Of these symptoms, few depend on specific neuroendocrine changes attributed to pregnancy. Amenorrhea can appear in 38% of hospitalized female patients awaiting ECT; the normal menstrual cycle recovers upon the remission of depression. b Galactorrhea and even regular lactation can result from chronic breast stimulation, less commonly from a constant irritative 320
focus such as a scar and/or inflammatory area at or near the breast, and from drugs, most commonly neuroleptics. I Medical Differential Diagnosis and Exclusions Once pregnancy and its complications have been excluded by an examination, imaging (i.e., radiographic and ultrasonic), and hormonal markers (i.e., beta-HCG, LH, FSH, and prolactin), as well as the early phases of menopause, drug side effects, morbid obesity, ascites, ovarian or uterine tumors and central nervous system tumors, syndromes of acyclic gonadotropin release, and abnormal ovarian folliculogenesis remain.? No single neurophysiologic pattern has been demonstrated; in separate cases, however, there have been elevations in prolactin, depressed levels of FSH, and occasionally persistent corpus luteum. In addition to prolactin, growth hormone secretion is also under dopaminergic control.' Pseudopregnancy manifested by physical and behavioral changes has been demonstrated in dogs, horses, rabbits, and rats along with evidence of a persistent corpus luteum; this structure may be involved in some human cases, possibly from increased prolactin and/or elevated secretion. 6 Pseudocyesis has been compared to galactorrhea-amenorrhea hyperprolactinemia syndrome (GAHS) due to pituitary prolactinoma; GAHS may begin spontaneously, after oral contraceptive use, or postpartum. Resolution of amenorrhea occurred in 7 of 22 patients, and resolution of galactorrhea occurred in 6 of 19 patients spontaneously. The initial prolactin levels, six times the upper limit of normal, declined by 30% over the study.K Most women diagnosed with GAHS do not believe they are pregnant, which makes the association less clear. On the other hand, a few cases in which GAHS has responded to psychological intervention, particularly for depression, have been recorded. 9 Stein-Leventhal polycystic ovarian disease, due to factors including inappropriate estrogen feedback and dopaminergic dysfunction, causes hypothalamic-pituitaryPSYCHOSOMATICS
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ovarian axis asynchrony and anovulatory amenorrhea. Although the hormonal profiles resemble pseudocyesis, the belief in pregnancy is not present. 7 Nosologic Status Psychological symptoms in pseudocyesis are poorly recorded, and most researchers seem to minimize the extent of psychopathology to make it fit the model of psychosomatic illness and psychodynamic theories. This may be due in part to the lack of follow-up studies. Because of the diverse nature of the underlying causes, the diagnosis borders phenomenologically on other disorders. Often patients with the clinical diagnosis of incomplete or spontaneous abortion have no products of conception after curettage; fantasies of pregnancy or its symptoms and signs have appeared months or years after postpartum sterilization in 80% of 190 subjects (adjustment disorders).lo The bulk of the cases in Bivin and Klinger4 suggest conversion disorder (hysteria) as a mechanism for most of the symptoms aside from the amenorrhea and galactorrhea, and the conflicting wish for and fear of pregnancy seems to accord with the criteria.4 The pregnant role provides an accessible, dependent, vulnerable social role that will evoke caring and attention from others and a measure of self-esteem. (This seems to be the case with one of the fictional characters in a story by LB. Singer, Enemies, a Love Story, recently made into a movie.) The mimicry of pregnancy can be modeled from a variety of sources. I A series from South Africa notes the custom of "Iobala," in which the bridegroom's family makes a payment to the bride's on fulfillment of the marriage contract by proof of pregnancy; as a result, there is an intense desire to be fertile or have children. II In addition, the issue of mimicry compares with the couvade syndrome,12 in which husbands experience some of the symptoms of pregnancy and/or delivery. The two forms found in traditional societies are the pseudomaternal (simulation of childbirth) and the dietetic (postnatal dietary restrictions) couvade. The most common sympVOLUME 33· NUMBER 3· SUMMER 1992
toms are gastrointestinal (e.g., indigestion or decreased appetite, diarrhea or constipation) and pains (e.g., aches, toothache, backache). A significantly larger number of expectant fathers (15%)13 suffered from one or more symptoms with a peak incidence at 3 months, with onethird clearing before labor began. Apart from anxiety, there was a significant relationship between the occurrence of physical symptoms and other psychiatric symptoms (i.e., depression, insomnia, irritability, weakness, headaches, stuttering, and tension).13 Men with couvade doubled their visits to physicians and had an increased rate of prescriptions. 12 Some subjects probably represent hypochondriasis in their excessive interpretation of physiologic symptoms. Most authors have mentioned the presence of anxiety or depression in the genesis of pseudocyesis. Bivin and Klinger4 discuss eight cases with depression of various degrees; negative attitudes (i.e., grief, grief and hysteria, grief and indignation, and indignation) were found in 34 (8%) of the 444 cases. 4 It has been hypothesized that the mood disorder alters the function of hypothalamic catecholaminergic areas, initiating and maintaining the syndrome. ' ·6 It is noteworthy that the duration of the natural course of an endogenous depression is between 9 and 12 months. Two of 47 patients of childbearing age who committed suicide had pseudocyesis (Hedda Gabler syndrome).'4 In the series of 444 patients, only 6 instances of "delusions" are noted without further elaboration in the text. 4 Psychotic depression '2 and mania may present with the disorder (major mood disorder). Rudden et aI. 1s noted erotically related beliefs (i.e., sexual pursuit, jealousy, impending marriage, pregnancy, rape, or venereal disease) in one-third of the women examined. They found higher depression scores among the women despite an equivalent number of diagnoses of major mood disorders. Again, more women had schizoaffective disorder or atypical psychosis with a greater number of sexual or family-related precipitants. IS Even in the comparatively uncommon association of pseudocyesis with Capgras syndrome, mood disorders predominate. ' 6-'9 321
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De Clerambault recorded the belief in an indefinitely prolonged (often several years) pregnancy among Moroccan women cal1ed "raqed," meaning "the sleeper." Its development seemed to be initiated by vague abdominal sensations and mild elation ("I' optimisme"). 20 This unpublished note suggested a link with the "psychoses passionnel1es" as the delusional content is limited to one theme ("en secteur") similar to the delusional disorders. More general1y psychotic patients with schizophrenia have also been recorded. 19 Of a series of patients with delusions due to traumatic brain injury, three had pseudocyesis; both women had diffuse brain swel1ing on CT, one had hypomania, visual and auditory hal1ucinations, and exhibitionism, and the other had visual hal1ucinations, geographic and personal reduplication, and exhibitionism. 21 The symptom of the denial of pregnancy forms an interesting counterpoint with diagnoses covering, one could expect, the same territory. Although more often reported as a curiosity in newspapers, it tragical1y forms the background for neonaticide. 22 - 24 Finally, the claim of pregnancy might be the product of deception either on the basis of a factitious disorder or malingering. Although the hypothesized changes in dopaminergic function in severe depression have been noted earlier, there are too few cases with organic antecedents to assign a potential mechanism. In these cases, a mood disorder with psychosis (one with depression, six with manic or mixed bipolar states) was present, but only in a minority of the reports. Three of the seven patients and three of the six manic or mixed patients in the series had right-sided cerebral injury, consistent with the literature. 25 It is difficult to fit the delusion into the disorders of body schema, such as asomatognosia (misperception of one's body) and any of its subdivisions, because they are unilateral. These syndromes appear not only with lesions of the parietal cortex, particularly on the right, but also with prefrontal association cortex and thalamic lesions. The most common area is the posterior temporoparietal cortex, in the borderzone of the middle and posterior cerebral artery circula322
tion. 26 Micro- and macrosomatognosia, in which parts of the body are perceived as abnormal1y smal1 or large, have been associated with paroxysmal disorders (e.g., epilepsy, migraine),26 and elaborate perceptual experiences of the body can occur with injury to the temporal lobes. Self-perception can be remarkably distorted with depersonalization, and it may form the core around which somatic delusions crystal1ize. 27 Depersonalization, or more properly desomatization when the perception of a body part is involved, can occur with a wide range of psychiatric disorders, including anxiety and depressive states of varying severity, which are important to the occurrence of the false belief in pregnancy. No clear anatomic localization has been determined, although the temporal and nondominant parietal lobes have been implicated. Similarly, at this point, no neuroanatomic substrate can be linked with pseudocyesis, except in terms of the underlying psychiatric disorder. Treatment Treatment depends on an accurate diagnosis of the underlying il1ness and the appropriate intervention. The patients do not respond to the mere information that they are not pregnant. Often, they will go from physician to physician seeking confirmation of the false beliefs and being distressed, unwilling to believe or unhappy to learn the truth. They tend to be resistant to psychological or psychiatric treatment, although those who accept it may make a good adjustment. Hormonal manipulation with progestational agents (medroxyprogesterone acetate, 10 mg), the use of imaging techniques such as ultrasound, or curettage may accompany the psychological interventions that may attempt to find a face-saving solution, deal with other losses, or resolve conflicts. \ CONCLUSION The syndrome of pseudocyesis does not appear to be a single, coherent entity. The physical symptoms can be assigned to a number of PSYCHOSOMATICS
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causes, some local, some musculoskeletal, and some honnonaI. A number (e.g., amenorrhea, abdominal proptosis) may be found without the necessity for the false belief in pregnancy. It cannot be placed into a single diagnostic category and ranges over the entire spectrum of
psychopathology. The majority of cases fall into either conversion or mood disorders. This group with pseudocyesis arising in the context of a psychotic mood disorder demonstrated organic contributors to their state.
References I. O'Grady JP. Rosenthal M: Pseudocyesis: a modem perspective on an old disorder. Obstet Gynecol Sun' 44:500-511. 1989 2. Dunbar JF: Emotions and Bodily Changes. New York. Columbia University Press. 1946. p 346 3. Small GW: Pseudocyesis: an overview. Can J Psychiatry 31:452-457.1986 4. Bivin GO. Klinger MP: Pseudocyesis. Bloomington. Principia Press. 1937 5. Hardy JL. Coulombe M: Abdominal proptosis: a case report. Can J Psychiatry 32:310-311. 1987 6. Brown E. Barglow P: Pseudocyesis: a paradigm of psychophysiological interactions. Arch Gen Psychiatry 24:221-229.1964 7. Ayers JWT. Seiler JC: Neuroendocrine indices of depression in pseudocyesis: a case report. J Reprod Med 29:67-70. 1984 8. Koppelman MCS. Jaffe MJ, Reith KG, et al: Hyperprolactinemia, amenorrhea and galactorrhea: a retrospective assessment of 25 cases. Ann Intern Med 100:115-121. 1984 9. Cohen LM: A current perspective of pseudocyesis. Am J Psychiatry 139:1140-1144, 1982 10. Barglow P: Pseudocyesis and psychiatric sequelae of sterilization. Arch Gen Psychiatry II :571-580. 1964 II. Brenner BN: Pseudocyesis in blacks. S Afr Med J 50:1757-1759, 1976 12. Trethowan WH, Conlon MF: The couvade syndrome. Br J Psychiatry III :57--{j6. 1965 13. Lipkin M. Lamb GS: The couvade syndrome: an epidemiolgic study. Ann Intern Med 96:509-511, 1982 14. Goodwin J. Harris 0: Suicide in pregnancy: the Hedda Gabler syndrome. Suicide Life Threat Behal' 9: 105-115, 1979 15. Rudden M. Sweeney J. Frances A. et al: A comparison of delusional disorders. Am J Psychiatry 140:1575--
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1578.1983 16. Frey B. Maurel H. Spielmann JP: Sur une observation d'illusion de sosie. Ann Med Psychol (Paris) 114:891896, 1956 17. Green BH. Birchall EW: Erotomania and Capgras syndrome in bipolar affective disorders. British Journal of Clinical and Social Psychiatry 6:51-53. 1988 18. Barton JL, Barton ES: Misidentification syndromes and sexuality, in "The Delusional Misidentification Syndromes." in Biblioteca Psychiatrica 164. edited by Christodoulou GN. Basel, Karger, 1986. pp 105-120 19. Mortimer A, Banbery J: Pseudocyesis preceding psychosis. Br J Psychiatry 152:562-565, 1988 20. de Clerambault G: A propos des delires de grossesse, importance des paresthesies. ('idee de grossesse prolongee chez les arabes (1922), in Oeul're Psychiatrique. edited by Fretet J. Paris. Presses Universitaires de France. 1942, pp 731-732 21. Levin HS, Benton AL. Grossman RG: Neurobehavioral Consequences ofClosed Head Injury. New York, Oxford University Press. 1982 22. Green CM, Manohar SV: Neonaticide and hysterical denial of pregnancy. Br J Psychiatry 156: 121-123,1990 23. Saunders E: Neonaticides following "secret" pregnancies: seven case reports. Public Health Rep 104:368372. 1989 24. Finnegan P, McKinstry E. Erlick Robinson G: Denial of pregnancy and childbirth. Can J Psychiatry 27:672-
STEPHEN F. SIGNER, M.D., ROBERT
P.
WEINSTEIN, M.D.
A. MUNOZ, M.D., J. FERNANDO BAYARDO, M.D. MARK R. KATZ, M.D., LAURENCE R. SABEN, M.D.
RODRIGO
Pseudocyesis. the delusion of pre~nancy. has had an uncertain nosolo~y. primarily because of the concentration on the content of the beliefs and lack of interest in the underlyin~ phenomenolo~y. Six patients with a major mood disorder caused by cerebral dysfunction are presented in this article. The delusion is reviewed with respect to the entities it overlaps. and the clinical manifestations are related to the mood disorders. Although no clear neuroanatomic localization was possible with this ~roup of patients. there may be some association with desomatization caused by parietal lobe dysfunction.
A
lthough a large number of case reports from the past have been accumulated, pseudocyesis has become uncommon in industrialized societies with the exception of a large series from West and South Africa. Because of the importance of determination of succession in many cultures, few delusions have shown such a clear influence on the course of history (Mary Tudor, Natalie of Serbia). The modem term, coined by Good in 1823 from the Greek root words for "false pregnancy," has replaced the variants in the older literature. I Pseudocyesis has been defined as a state in which a Received June 3. 1991; revised August 30. 1991; accepted September 6. 1991. From the Depanment of Psychially, University of California. San Diego. Address reprint requests to Dr. Signer, Psychiatric Centers at San Diego, 15725 Pomerado Road. Suite 206, Poway (San Diego), CA 92064. Copyright © 1992 The Academy of Psychosomatic Medicine.
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woman believes herself to be pregnant, developing the commonly occurring symptoms and signs (i.e., amenorrhea, gradual abdominal enlargement, breast changes, fetal movement, morning nausea and vomiting, and weight gain) in the absence of actual pregnancy. Although defined as a false belief (delusion) in that the patient insists on the reality of the pregnancy despite evidence to the contrary, pseudocyesis has been considered a conversion symptom that is exclusive of pseudopregnancy (medical illness), simulated pregnancy (malingering), or hallucinatory pregnancy (psychosis).2 As such, pseudocyesis is regarded as a paradigm of psychosomatic disorders even though the phenomenology has been quite variably and poorly recorded. The traditional emphasis on presumed psychodynamic content and conflicts ignores and downplays the more modem interest in the importance of phenomenologic features in determining the nosology. At this point, it is PSYCHOSOMATICS
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inappropriate to allow theoretical considerations to interfere with the explorations of the boundaries of a distinct syndrome. This article presents six new cases of the delusion of pregnancy caused by brain injury in the context of a mood disorder (organic mood disorder) and reviews the other disorders in which this false belief can occur. Case Reports Case 1. Ms. A.• a 39-year-old. right-handed separated woman of mixed North American Indian and French-Canadian descent. had three children (22. 16. and 7 years of age). She presented to her general doctor with 4 months of amenorrhea. increasing abdominal pain. and the sensation of internal movement. and he confirmed her belief that she was pregnant. After a negative pregnancy test. a pelvic ultrasound was done that showed a normal nongravid abdomen. When Ms. A. was told she was not pregnant, she became grossly agitated. believing there was some sort of monster or worms inside her. On admission. she complained of numbness all over her body, depressed mood. sleep disturbance. and hallucinations commanding her to harm herself. Ms. A.·s mother had recurrent depression and other family members had alcoholism. Ms. A. had had a brutal childhood with physical and sexual abuse. She became pregnant after a rape at the age of 17 years. and the child was raised by her mother; a second child. born when she was 23 years old. was given to the care of the Children's Aid Society (CAS). She separated from her alcoholic husband 3 years after the birth of her last child and found the repeated contact with him very stressful. One month before the onset of symptoms. the supervision of a CAS social worker, whom she liked, ended. On examination there was no galactorrhea and her uterus was normal. Urine pregnancy. betahuman chorionic gonadotropin (HCG). and dexamethasone suppression tests were negative. One measure of serum prolactin was elevated at 36 ~g/L. Thyroid function tests were normal. The brain cr scan was normal; the EEG showed bilateral mild slow wave disturbances. On the Wechsler Adult Intelligence Scale-Revised (WAIS-R), the verbal subscale was 53. the performance subscale was 72. and the full scale was 58; the Wechsler Memory Quotient was 52. Her educational level was below VOLUME 33· NUMBER 3· SUMMER 1992
that of the 3rd grade in elementary school. She was treated with fluphenazine decanoate 50 mg im twice a month for a diagnosis of delusional disorder. Ms. A. was readmitted 6 weeks later with depressed mood; diurnal variation; crying; agitation; anorexia; initial. middle. and terminal insomnia; fatigue; loss of interest; social withdrawal; poor concentration; and homicidal and suicidal ideation. She complained again of the feelings of numbness and milky discharge from her right breast. although none could be expressed. but did not have the delusion of false pregnancy. At that point the diagnosis of unipolar depression was much clearer. and she responded to treatment with imipramine 125 mg qd and haloperidol 10 mg qd. Case 2. Ms. B.• a 29-year-old. right-handed twicemarried Hispanic woman, claimed to have had 30 children. mostly twins, after eight pregnancies. She believed that she was again pregnant with twins and was experiencing amenorrhea. abdominal protrusion. heartburn and nausea, weight gain. breast tenderness. and the sensation of movement; there were two episodes of false labor. Ms. B. achieved menarche at age 12 years with irregular cycles. having mild postpartum dysphoria. She said all the children were taken from her at birth. Her medical history was noncontributory aside from past binge drinking and drug abuse, including cannabis. narcotics, hallucinogens. psychostimulants, sedatives. and solvents. The patient alternated between periods of depression and elation. The depressions lasted 1-2 weeks with decreased appetite and weight. decreased energy, increased sleep. and psychomotor slowing. Hypomanic states were longer lasting. with increased activity, decreased sleep. and grandiosity. She had been hyperactive and destructive as a child; there were several hospitalizations for mania with grandiose delusions. Ms. B. was elated, with rapid. pressured speech and thought form marked by tangentiality. circumstantiality. and derailment. She had some unorganized ideas of surveillance. but denied erotic. grandiose, control, substitution. or other somatic delusions. The digit span was six forward but none in reverse; the reversal of other learned sequences was slowed. Her language function was normal. although verbal fluency was decreased; confabulation appeared in a test of short-term memory. The ReyOsterreith figure showed distortion on the right. segmental approach. with perseveration and fusion with previous form. A clock drawing showed perseveration to the number 19; the Luria hand se317
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quence demonstrated verbal-motion dissociation on the right. The motor examination showed rightsided drift. increased tone. and retlexes. The pregnancy test was negative. Treatment with lithium. tluphenazine. and amitriptyline stabilized her mood state. and the delusion disappeared. Case 3. Ms. c.. a 68-year-old. right-handed former clerk. claimed six previous marriages and seven children. She threatened others with knives and was preoccupied with cutting up objects. The patient was elated. with an increased level of activity. pressured rapid speech. derailment. tangentiality. and circumstantiality. Ms. C. denied having been depressed although some mixed affective symptoms were present; she characterized herself as a "super energy" person. Ms. C. claimed that a younger fundamentalist preacher had married her in May (erotomania) and that she was 2 months pregnant. She claimed morning sickness. increased weight. and breast fullness; she soon expected to feel movement and wanted a caesarean section. Menarche had occurred at age 13 years and she denied any menopausal symptoms. Ms. C.·s father was an "inventor" and alcoholic. as were many members of his family. She had diabetes mellitus. hypenension. and coronary anery disease; her medications included chlorpropamide. c1onidine. diltiazem. and nitroglycerin. to which lithium was added with control of the symptoms. Her attention was mildly impaired; language was normal although word list generation was markedly decreased; shon-term memory was impaired with confabulation; and Taylor figure showed a segmental strategy. with recall demonstrating a loss of form. The neurologic examination showed extinction of double simultaneous stimulation in the left visual field. decreased upward gaze. decreased saccades to the left. left facial weakness. and increased tone and grasp retlex on the left with distraction maneuvers. The CT scan demonstrated an infarction in the right basal ganglia and anterior limb of the internal capsule and frontal lobe. The mood cycles diminished with the use of lithium. and the delusions were controlled with thioridazine. Case 4. Ms. D.• a 27-year-old. right-handed woman. had prolonged intraoperative anoxia with subsequent coma at age 17 years. She had noted some right-handed weakness and difficulty in writing. Ms. D. had been hospitalized several times. the last for agitation and persecutory delusions. with auditory hallucinations and thought broadcasting. In 3111
addition to some periods of depression. the patient had episodes of elation. with promiscuity and impulsive overspending. The patient believed herself to be pregnant without intercourse. claiming amenorrhea. breast fullness and enlargement. increased appetite with a desire for healthful food. and firmness across her abdomen. Menarche had been achieved at age 12 years. premenstrual affective changes were denied. and she claimed previous pregnancy without intercourse lasting 10 months and delivering a "goldencolored" boy. The pregnancy test was negative. and the family history was noncontributory. She had abused cocaine during two extended periods. Two seizures of unknown phenomenology had occurred. She was treated with lithium. carbamazepine. and thiothixene. with poor response either of the elated mood or symptoms of pseudocyesis. Her attention was normal although she performed both parts of the Trail-Making Test below the 50th percentile; language was normal and there was a marked disparity between the word list generation for concrete objects (animals 17) and letters (using F. A. and S. total = 25). Confabulation appeared on testing shortterm memory. which improved with repetition and cuing; writing showed micrographia of the frontal lobe variety; perseveration was present in drawings; and the Rey-Osterreith figure showed loss of detail and perseveration on recall. The neurologic examination demonstrated difficulty moving the eyes past the midline to the left. decreased left optokinetic nystagmus; decreased strength on the left. increased tone on the left with distraction maneuvers. and increased reflexes on the left. Case 5. Ms. E.. a 43-year-old. right-handed housewife. was admitted in an agitated psychotic state after several weeks of deterioration. She had been hospitalized in similar circumstances in the past. She had had episodes of depression. withdrawal. and poor self-care as well as others of increased activity. frequently harassing calls or letters to others. and increased spending. In addition to ideas that others could control her mind and were accusing her of stealing. Ms. E. believed that she was pregnant because of amenhorrhea. The patient thought her appetite decreased with occasional vomiting. increased weight. abdominal firmness. increased breast size with galactorrhea. and fetal movements. The patient was born I month premature. delivered by high forceps. and at 6 months had a viral infection that caused paralysis of the palate. tlatPSYCHOSOMATICS
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tened affect, slow development of developmental milestones, and learning and speech problems. She had diabetes mellitus, asthma, and hypothyroidism. Ms. E. did not have children, although she had had several miscarriages and a "missed" abortion led to one hospitalization. She had a loaded pedigree for affective disorders. The patient showed a psychomotor retarded parkinsonian appearance. Attention and memory were impaired with considerable perseveration. Right-sided neglect was present on the Taylor figure. Ms. E. demonstrated an increased left palpebral fissure, dysarthria, left-sided clumsiness, increased left-sided reflexes, a small left hand, bilateral grasp reflex, and a positive glabellar tap. Psychological testing showed full-scale IQ to be 69 (verbal 62, performance 67). The thyroid-stimulating hormone was 13.9. The EEG showed diffuse slowing and the CT scan prominent cerebral atrophy. The delusions disappeared with the use of neuroleptics, which had to be stopped due to akathisia and secondary parkinsonian symptoms. Case 6. Ms. E, a 59-year-old, right-handed woman with four children, was admitted with a psychosis characterized by depression, loss of appetite, insomnia, lethargy, suicidal ideation, auditory hallucinations. and persecutory delusions. Over the past 30 years she had had over to psychiatric hospitalizations for both depressive and manic episodes. requiring treatment with electroconvulsive therapy (ECT). neuroleptics, lithium, and carbamazepine at various times. Ms. E had been prescribed lithium and fluphenazine. pilocarpine for glaucoma. digoxin for cardiac arrhythmia, and theophylline for chronic obstructive lung disease. In addition to hypertension. the patient had small-cell carcinoma of the lung treated with radiotherapy and chemotherapy. Ms. E had the recent onset of the belief that she was 9 months pregnant with weight gain of 10 Ibs., abdominal swelling, and the sensation of fetal movements and false labor; she was unsure whether to attribute amenorrhea of 8 years' duration to the "pregnancy" or to "change of life." The patient was irritable and uncooperative. Her affect was somewhat blunted and mood depressed, and hallucinations were not endorsed. The patient was inattentive with psychomotor slowing, and mild deficits in language and memory were present; speech was aprosodic. The neurologic examination did not demonstrate lateralizing signs. Her full-scale WAIS-R was 83. The CTshowed multiple small areas of infarction in the brain stem VOLUME 33· NUMBER 3· SUMMER 1992
and frontal lobes. The pregnancy test was negative; follicle-stimulating hormone (FSH) was 8 mIU/ml, luteinizing hormone (LH) was I mIU/ml, and prolactin was 44 mIU/ml. The mood changes and psychosis remitted with lithium, haloperidol, and clonazepam.
DISCUSSION All of the patients exhibited the delusion of pregnancy with the appropriate physical symptoms in the setting of a psychosis associated with a major mood disorder due to identifiable organic deficits. All had abdominal symptoms including the sensation of fetal movement. Ms. A., who evolved more neurovegetative symptoms of depression over time, was moderately mentally retarded with diffuse EEG changes. Ms. B. exhibited a course of alternating depression and elation with the presence of delusion in the latter phase; evidence of left-hemispheric injury was apparent on the cognitive and neurologic examinations. Ms. C. had a right basal ganglia infarction. Ms. D. had an anoxic encephalopathy with predominantly right hemispheric signs. Ms. E. had several causes for brain injury in the first years of life; there were predominantly right-hemispheric signs present. Ms. F., while ill with schizoaffective disorder, had the new onset of the delusion with a dementia due to white matter infarction from embolic disease. In this group with antecedent organic features, the association seems to be with a severe mood disorder with psychosis, rather than with any lateralized (three right-. one lefthemisphere; two diffuse injury) neuropsychological features, although distortion of body image may be more common with right-hemispheric injury. Aside from Ms. A., who showed a major depression with psychosis, all the cases demonstrated bipolar mood disorders with psychosis. Demographic Characteristics There are no clear demographic or sociocultural indicators for the population at risk. Subjects have ranged in age from 5 to 79 years, 319
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with an average of 33 years; 73% were between 15 and 39 years; and 17% were postmenopausal. Most of the patients were married, 37% had been pregnant previously, and 5% had recurrent pseudocyesis. The symptoms had lasted 9 months in 43%,1.3.4 rarely persisting for 1-2 years. Clinical Manifestations The most common symptoms and signs include the following (from a combined series):1.3 menstrual abnormalities (oligomenorrhea, and uncommonly amenorrhea; 69%), gradual abdominal enlargement (68%), and the sensation offetal movement (63%). A history of infertility (59%), breast changes (enlargement, tenderness, pigmentation; 59%), weight gain (44%), galactorrhea (42%), and cervical changes (40%) have also been noted.u.4 Morning sickness may appear before the first missed menstrual period and the quickening occurs earlier than in a real pregnancy. The abdomen is usually rounded with uniform distension and tense, rubbery tone and weight gain is usually greater than normal. The umbilicus is inverted, the cervix firm, the uterus not enlarged, and no fetal parts are palpable. The abdominal distention, due probably to changes in tone of the diaphragm and abdominal muscles, lordosis, and possibly omental and abdominal fat, and bowel distention, usually disappears with steady pressure, Bargen's maneuver, anesthesia, or muscle relaxants. 4 .5 False labor occurred in I% of a group of modem series' and 30% of the historical group. 4 Fetal movements, atypical for their intensity, period of onset, or pattern, may be so violent as to be socially disruptive and presumably are due to abdominal muscle contractions. I Of these symptoms, few depend on specific neuroendocrine changes attributed to pregnancy. Amenorrhea can appear in 38% of hospitalized female patients awaiting ECT; the normal menstrual cycle recovers upon the remission of depression. b Galactorrhea and even regular lactation can result from chronic breast stimulation, less commonly from a constant irritative 320
focus such as a scar and/or inflammatory area at or near the breast, and from drugs, most commonly neuroleptics. I Medical Differential Diagnosis and Exclusions Once pregnancy and its complications have been excluded by an examination, imaging (i.e., radiographic and ultrasonic), and hormonal markers (i.e., beta-HCG, LH, FSH, and prolactin), as well as the early phases of menopause, drug side effects, morbid obesity, ascites, ovarian or uterine tumors and central nervous system tumors, syndromes of acyclic gonadotropin release, and abnormal ovarian folliculogenesis remain.? No single neurophysiologic pattern has been demonstrated; in separate cases, however, there have been elevations in prolactin, depressed levels of FSH, and occasionally persistent corpus luteum. In addition to prolactin, growth hormone secretion is also under dopaminergic control.' Pseudopregnancy manifested by physical and behavioral changes has been demonstrated in dogs, horses, rabbits, and rats along with evidence of a persistent corpus luteum; this structure may be involved in some human cases, possibly from increased prolactin and/or elevated secretion. 6 Pseudocyesis has been compared to galactorrhea-amenorrhea hyperprolactinemia syndrome (GAHS) due to pituitary prolactinoma; GAHS may begin spontaneously, after oral contraceptive use, or postpartum. Resolution of amenorrhea occurred in 7 of 22 patients, and resolution of galactorrhea occurred in 6 of 19 patients spontaneously. The initial prolactin levels, six times the upper limit of normal, declined by 30% over the study.K Most women diagnosed with GAHS do not believe they are pregnant, which makes the association less clear. On the other hand, a few cases in which GAHS has responded to psychological intervention, particularly for depression, have been recorded. 9 Stein-Leventhal polycystic ovarian disease, due to factors including inappropriate estrogen feedback and dopaminergic dysfunction, causes hypothalamic-pituitaryPSYCHOSOMATICS
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ovarian axis asynchrony and anovulatory amenorrhea. Although the hormonal profiles resemble pseudocyesis, the belief in pregnancy is not present. 7 Nosologic Status Psychological symptoms in pseudocyesis are poorly recorded, and most researchers seem to minimize the extent of psychopathology to make it fit the model of psychosomatic illness and psychodynamic theories. This may be due in part to the lack of follow-up studies. Because of the diverse nature of the underlying causes, the diagnosis borders phenomenologically on other disorders. Often patients with the clinical diagnosis of incomplete or spontaneous abortion have no products of conception after curettage; fantasies of pregnancy or its symptoms and signs have appeared months or years after postpartum sterilization in 80% of 190 subjects (adjustment disorders).lo The bulk of the cases in Bivin and Klinger4 suggest conversion disorder (hysteria) as a mechanism for most of the symptoms aside from the amenorrhea and galactorrhea, and the conflicting wish for and fear of pregnancy seems to accord with the criteria.4 The pregnant role provides an accessible, dependent, vulnerable social role that will evoke caring and attention from others and a measure of self-esteem. (This seems to be the case with one of the fictional characters in a story by LB. Singer, Enemies, a Love Story, recently made into a movie.) The mimicry of pregnancy can be modeled from a variety of sources. I A series from South Africa notes the custom of "Iobala," in which the bridegroom's family makes a payment to the bride's on fulfillment of the marriage contract by proof of pregnancy; as a result, there is an intense desire to be fertile or have children. II In addition, the issue of mimicry compares with the couvade syndrome,12 in which husbands experience some of the symptoms of pregnancy and/or delivery. The two forms found in traditional societies are the pseudomaternal (simulation of childbirth) and the dietetic (postnatal dietary restrictions) couvade. The most common sympVOLUME 33· NUMBER 3· SUMMER 1992
toms are gastrointestinal (e.g., indigestion or decreased appetite, diarrhea or constipation) and pains (e.g., aches, toothache, backache). A significantly larger number of expectant fathers (15%)13 suffered from one or more symptoms with a peak incidence at 3 months, with onethird clearing before labor began. Apart from anxiety, there was a significant relationship between the occurrence of physical symptoms and other psychiatric symptoms (i.e., depression, insomnia, irritability, weakness, headaches, stuttering, and tension).13 Men with couvade doubled their visits to physicians and had an increased rate of prescriptions. 12 Some subjects probably represent hypochondriasis in their excessive interpretation of physiologic symptoms. Most authors have mentioned the presence of anxiety or depression in the genesis of pseudocyesis. Bivin and Klinger4 discuss eight cases with depression of various degrees; negative attitudes (i.e., grief, grief and hysteria, grief and indignation, and indignation) were found in 34 (8%) of the 444 cases. 4 It has been hypothesized that the mood disorder alters the function of hypothalamic catecholaminergic areas, initiating and maintaining the syndrome. ' ·6 It is noteworthy that the duration of the natural course of an endogenous depression is between 9 and 12 months. Two of 47 patients of childbearing age who committed suicide had pseudocyesis (Hedda Gabler syndrome).'4 In the series of 444 patients, only 6 instances of "delusions" are noted without further elaboration in the text. 4 Psychotic depression '2 and mania may present with the disorder (major mood disorder). Rudden et aI. 1s noted erotically related beliefs (i.e., sexual pursuit, jealousy, impending marriage, pregnancy, rape, or venereal disease) in one-third of the women examined. They found higher depression scores among the women despite an equivalent number of diagnoses of major mood disorders. Again, more women had schizoaffective disorder or atypical psychosis with a greater number of sexual or family-related precipitants. IS Even in the comparatively uncommon association of pseudocyesis with Capgras syndrome, mood disorders predominate. ' 6-'9 321
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De Clerambault recorded the belief in an indefinitely prolonged (often several years) pregnancy among Moroccan women cal1ed "raqed," meaning "the sleeper." Its development seemed to be initiated by vague abdominal sensations and mild elation ("I' optimisme"). 20 This unpublished note suggested a link with the "psychoses passionnel1es" as the delusional content is limited to one theme ("en secteur") similar to the delusional disorders. More general1y psychotic patients with schizophrenia have also been recorded. 19 Of a series of patients with delusions due to traumatic brain injury, three had pseudocyesis; both women had diffuse brain swel1ing on CT, one had hypomania, visual and auditory hal1ucinations, and exhibitionism, and the other had visual hal1ucinations, geographic and personal reduplication, and exhibitionism. 21 The symptom of the denial of pregnancy forms an interesting counterpoint with diagnoses covering, one could expect, the same territory. Although more often reported as a curiosity in newspapers, it tragical1y forms the background for neonaticide. 22 - 24 Finally, the claim of pregnancy might be the product of deception either on the basis of a factitious disorder or malingering. Although the hypothesized changes in dopaminergic function in severe depression have been noted earlier, there are too few cases with organic antecedents to assign a potential mechanism. In these cases, a mood disorder with psychosis (one with depression, six with manic or mixed bipolar states) was present, but only in a minority of the reports. Three of the seven patients and three of the six manic or mixed patients in the series had right-sided cerebral injury, consistent with the literature. 25 It is difficult to fit the delusion into the disorders of body schema, such as asomatognosia (misperception of one's body) and any of its subdivisions, because they are unilateral. These syndromes appear not only with lesions of the parietal cortex, particularly on the right, but also with prefrontal association cortex and thalamic lesions. The most common area is the posterior temporoparietal cortex, in the borderzone of the middle and posterior cerebral artery circula322
tion. 26 Micro- and macrosomatognosia, in which parts of the body are perceived as abnormal1y smal1 or large, have been associated with paroxysmal disorders (e.g., epilepsy, migraine),26 and elaborate perceptual experiences of the body can occur with injury to the temporal lobes. Self-perception can be remarkably distorted with depersonalization, and it may form the core around which somatic delusions crystal1ize. 27 Depersonalization, or more properly desomatization when the perception of a body part is involved, can occur with a wide range of psychiatric disorders, including anxiety and depressive states of varying severity, which are important to the occurrence of the false belief in pregnancy. No clear anatomic localization has been determined, although the temporal and nondominant parietal lobes have been implicated. Similarly, at this point, no neuroanatomic substrate can be linked with pseudocyesis, except in terms of the underlying psychiatric disorder. Treatment Treatment depends on an accurate diagnosis of the underlying il1ness and the appropriate intervention. The patients do not respond to the mere information that they are not pregnant. Often, they will go from physician to physician seeking confirmation of the false beliefs and being distressed, unwilling to believe or unhappy to learn the truth. They tend to be resistant to psychological or psychiatric treatment, although those who accept it may make a good adjustment. Hormonal manipulation with progestational agents (medroxyprogesterone acetate, 10 mg), the use of imaging techniques such as ultrasound, or curettage may accompany the psychological interventions that may attempt to find a face-saving solution, deal with other losses, or resolve conflicts. \ CONCLUSION The syndrome of pseudocyesis does not appear to be a single, coherent entity. The physical symptoms can be assigned to a number of PSYCHOSOMATICS
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causes, some local, some musculoskeletal, and some honnonaI. A number (e.g., amenorrhea, abdominal proptosis) may be found without the necessity for the false belief in pregnancy. It cannot be placed into a single diagnostic category and ranges over the entire spectrum of
psychopathology. The majority of cases fall into either conversion or mood disorders. This group with pseudocyesis arising in the context of a psychotic mood disorder demonstrated organic contributors to their state.
References I. O'Grady JP. Rosenthal M: Pseudocyesis: a modem perspective on an old disorder. Obstet Gynecol Sun' 44:500-511. 1989 2. Dunbar JF: Emotions and Bodily Changes. New York. Columbia University Press. 1946. p 346 3. Small GW: Pseudocyesis: an overview. Can J Psychiatry 31:452-457.1986 4. Bivin GO. Klinger MP: Pseudocyesis. Bloomington. Principia Press. 1937 5. Hardy JL. Coulombe M: Abdominal proptosis: a case report. Can J Psychiatry 32:310-311. 1987 6. Brown E. Barglow P: Pseudocyesis: a paradigm of psychophysiological interactions. Arch Gen Psychiatry 24:221-229.1964 7. Ayers JWT. Seiler JC: Neuroendocrine indices of depression in pseudocyesis: a case report. J Reprod Med 29:67-70. 1984 8. Koppelman MCS. Jaffe MJ, Reith KG, et al: Hyperprolactinemia, amenorrhea and galactorrhea: a retrospective assessment of 25 cases. Ann Intern Med 100:115-121. 1984 9. Cohen LM: A current perspective of pseudocyesis. Am J Psychiatry 139:1140-1144, 1982 10. Barglow P: Pseudocyesis and psychiatric sequelae of sterilization. Arch Gen Psychiatry II :571-580. 1964 II. Brenner BN: Pseudocyesis in blacks. S Afr Med J 50:1757-1759, 1976 12. Trethowan WH, Conlon MF: The couvade syndrome. Br J Psychiatry III :57--{j6. 1965 13. Lipkin M. Lamb GS: The couvade syndrome: an epidemiolgic study. Ann Intern Med 96:509-511, 1982 14. Goodwin J. Harris 0: Suicide in pregnancy: the Hedda Gabler syndrome. Suicide Life Threat Behal' 9: 105-115, 1979 15. Rudden M. Sweeney J. Frances A. et al: A comparison of delusional disorders. Am J Psychiatry 140:1575--
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1578.1983 16. Frey B. Maurel H. Spielmann JP: Sur une observation d'illusion de sosie. Ann Med Psychol (Paris) 114:891896, 1956 17. Green BH. Birchall EW: Erotomania and Capgras syndrome in bipolar affective disorders. British Journal of Clinical and Social Psychiatry 6:51-53. 1988 18. Barton JL, Barton ES: Misidentification syndromes and sexuality, in "The Delusional Misidentification Syndromes." in Biblioteca Psychiatrica 164. edited by Christodoulou GN. Basel, Karger, 1986. pp 105-120 19. Mortimer A, Banbery J: Pseudocyesis preceding psychosis. Br J Psychiatry 152:562-565, 1988 20. de Clerambault G: A propos des delires de grossesse, importance des paresthesies. ('idee de grossesse prolongee chez les arabes (1922), in Oeul're Psychiatrique. edited by Fretet J. Paris. Presses Universitaires de France. 1942, pp 731-732 21. Levin HS, Benton AL. Grossman RG: Neurobehavioral Consequences ofClosed Head Injury. New York, Oxford University Press. 1982 22. Green CM, Manohar SV: Neonaticide and hysterical denial of pregnancy. Br J Psychiatry 156: 121-123,1990 23. Saunders E: Neonaticides following "secret" pregnancies: seven case reports. Public Health Rep 104:368372. 1989 24. Finnegan P, McKinstry E. Erlick Robinson G: Denial of pregnancy and childbirth. Can J Psychiatry 27:672-
