Delusion of Pregnancy

Abstract

Background:

Delusions can complicate practically all brain disorders. They may be dramatic and bizarre. An example is the so-called delusion of pregnancy.

Objective:

To identify the characteristic of a psychotic symptom, the phenomenon of delusion of pregnancy, in the context of dementia.

Method:

MEDLINE and Google Scholar searches were conducted for relevant articles, chapters, and books published before 2014. Search terms used included delusion of pregnancy, uncommon presentation, behavioral and psychological symptoms, dementia, Alzheimer’s disease, and frontotemporal dementia (FTD). Publications found through this indexed search were reviewed for further relevant references. We included case reports that highlight the relationship and overlap between dementia presenting as schizophrenia-like psychosis and schizophrenia.

Results:

Literature on delusion of pregnancy in the course of dementia consists mostly of case reports and small samples of patients.

Conclusion:

Psychotic phenomena such as delusion of pregnancy may be a feature in some cases of dementia. If this bizarre features of dementia appears as early presentation of FTD whose usual onset is in the presenium, it may be mistaken for schizophrenia.

Introduction

Delusions are fixed beliefs that are not amenable to change in light of conflicting evidence: their content may include a variety of themes (eg, persecutory, referential, somatic, religious, and grandiose). ¹ It is sometimes difficult, if not impossible, to establish the nonveridical nature of a belief. ² Delusion can complicate practically all neurological disorders, including epilepsy, cerebrovascular disorders, brain trauma, and brain tumors; its pathophysiology is uncertain and probably differs from one disorder to another. ³ It is a common, disabling, and persistent symptom in the course of dementia. ^4,5 Delusional symptoms are also described in Alois Alzheimer’s first case report of dementia. ⁶ They may be dramatic and bizarre. ⁷ An example is the so-called delusion of pregnancy.

Delusion of Pregnancy

The delusion of pregnancy is the condition of believing one is pregnant despite factual evidence to the contrary. According to Manjunatha et al, ⁸ it is reported among delusional procreation syndrome (DPS) that consists of sequential delusions in every possible stage of procreation such as having spouse/partner, getting pregnant, having delivered a child (labor and childbirth), becoming parents/grandparents and so on. ⁹ It should be distinguished from pseudocyesis (term coined in 1970 by Barglow and Brown) ¹⁰ that is a state when a nonpregnant woman has a false belief that she is pregnant and presents marked bodily signs of pregnancy. A person with delusion of pregnancy tends to believe that she is expecting a child, but this belief is not associated with bodily changes observed during pregnancy. ¹¹ Michael et al ¹² proposed to differentiate delusion of pregnancy from pseudopregnancy, Couvade syndrome, and simulated pregnancy. Pseudopregnancy is a condition triggered by organic factor such as endocrine tumor creating changes suggestive of pregnancy. ¹³ Couvade syndrome (sympathetic pregnancy) affects the male partners of pregnant women who experience a range of physical and psychological symptoms as expectant mothers with cessation of symptoms upon birth or shortly within the postpartum period. ¹⁴ Another concept to be considered is simulated pregnancy (when a woman admits to be pregnant, although she is aware that she is not), ¹⁵ actually described as malingering. According to American Psychiatric Association, the essential feature of malingering is the intentional production of false or grossly exaggerated physical or psychological symptoms, motivated by external incentives such as avoiding military duty, avoiding work, obtaining financial compensation, evading criminal prosecution, or obtaining drugs. ¹ Historically, the first documented case of delusional pregnancy was described by Esquirol at the turn of the 19th century in his treatise Des Maladies Mentales. ¹⁶ A single woman, 31 years of age, imagined herself to be impregnated by her botany teacher. She stopped eating, lost weight, and died 18 months later. It is nosologically nonspecific, occurring in a wide variety of organic states (posttraumatic epilepsy, ¹⁷ new variant of Creutzfeldt-Jakob disease, ¹⁸ and other organic brain syndromes), and functional psychoses, that is, psychotic conditions for which no organic lesions and no toxins have been consistently demonstrated (schizophrenia and mood disorders) ¹⁹ as well in medical conditions like drug-induced lactation. ²⁰ It has been reported both among male ^{21 -23} and female patients (also in postmenopausal women). ²⁴ The number of recorded instances of delusions of pregnancy in men seems to be very small, ²¹ and they have been occasionally reported as symptoms of a wide variety of psychotic states including schizophrenia, ²¹ schizoaffective disorders, ²¹ and bipolar mood disorders. ^{12 -22} Organic brain damage plays a strong part in the development of these delusional believes in men being reported in mental retardation, ²¹ postencephalitic syndrome, ²⁵ and general paresis. ²⁶ The coexistent appearance of delusions of pregnancy and infestation was reported in a male patient with posttraumatic epilepsy. ¹⁷ Also contact with pregnant women may produce the delusion in man. ²² Delusion of pregnancy attributed to sexual abuse in men has been reported. ²⁷ It was described in postmenopausal women, ²⁸ during postpartum period, ²⁹ in woman who had undergone hysterectomy, ²⁹ and in female with intense desire to become pregnant. ³⁰ Delusions of pregnancy associated with hyperprolactinemia were described. ^{31 -33} According to Levy et al, ³⁴ this association is biologically plausible: some similitude such as physiologic increase in prolactin during pregnancy (gestational signal) and similar symptoms to those during pregnancy, are strong argument in the onset of the delusional belief. However, the authors sustain that the interaction is more complex because not everyone with hyperprolactinemia will develop a delusion of pregnancy. Also neuroleptic therapy induced hyperprolactinemia with delusional ideas of being pregnant resolving as prolactin levels returned to normal. ³³ Cohen ³⁵ reported 2 cases of delusional pregnancy in patients with manifestations of Hashimoto’s thyroiditis (HT). The disruptive effect of thyroid disease on normal menstrual cycling is well recognized. According to the author, the amenorrhea or dysmenorrhea that may accompany thyroid disturbances in women could be misinterpreted as a pregnancy. Penta and Lasalvia ³⁶ reported a case of delusional pregnancy occurring in a young female with both paranoid schizophrenia and HT.

Etiology

It is highly doubtful that delusion of pregnancy can be explained by a single etiological factor. It can be triggered by organic factors without psychodynamic background or it can develop as an adaptation to stress induced by organic (eg, endocrine) and/or psychological factors. ³⁷ Coenesthopathological processes, in which combination of primary somatic sensations arising from the abdomen and other parts of the body due to central obesity ²³ and endocrinological changes, ³⁸ were misinterpreted as signs of pregnancy leading some patients to develop delusions of pregnancy. ³⁹ In the context of being cued to be vigilant for symptoms of pregnancy, a small amount of enteroceptor signal that is perceived to be in keeping with pregnancy may result in disproportionate emotional arousal, and a feedback loop may be established that keeps attention focused on these symptoms, resulting in an increasingly firm belief in the pregnancy. ³³ Cognitive theory hypothesizes that delusions arise from normal cognitive processes directed at explaining abnormal perceptual experiences ⁴⁰ influenced by premorbid values and beliefs and in the context of information-processing vulnerabilities. Sociocultural factors are not negligible in understanding believes about pregnancy. For example, in the Christian culture, pregnancy is regarded as a “blessed state” and it is surrounded with respect and acknowledgment, which may find its roots in the fertility cult of the archaic folks. ²⁹ Another example, a Muslim man can marry 4 times and hence can have 4 wives; given these social realities, the patient with delusional pregnancy may seek to prevent her husband from marrying another woman who might insist that her prospective husband divorce the patient. ¹⁹ Maher and Spitzer ⁴¹ explains that the patient is delusional because he or she actually experiences anomalies that demand explanation. The delusional explanation offers relief from puzzlement, and that relief works against abandonment of the explanation.

Delusion of Pregnancy and Dementia

Patients with delusion of pregnancy are described in the context of dementia. It has been documented in patients having Alzheimer’s disease (AD). Harland and Warner ²⁸ reported the case of 86-year-old widow who was admitted to hospital due to a progression of a dementing illness probably Alzheimer’s type. On admission, she had the delusion that she was pregnant and said her abdominal pain was due to the baby moving. She described her pregnancy as a miracle but had no other explanations. On treatment with trifluoperazine 1 mg twice a day, her delusion resolved within 3 days. The same authors ²⁸ referred to a 73-year-old widow with a 9-year history of progressive cognitive impairment due to dementia of probable Alzheimer’s type. She believed that she was pregnant. This lady also had abdominal distention due to obesity. The delusional symptom resolved after treatment with trifluoperazine 2 mg twice a day. Delusion of pregnancy has also been observed in vascular dementia. ²⁸ A 75-year-old widowed lady had an acute episode of verbal and physical aggression coinciding with a significant deterioration of her cognitive state. Over the next week, she developed the delusional belief that she was pregnant. She also believed she gave birth to a baby girl one morning recently. Some reports described delusion of pregnancy in frontotemporal dementia (FTD). It is the second most common form of dementia in those younger than 65 years, after AD. Clinically, FTD is characterized by behavioral and/or language dysfunction. A variety of psychiatric symptoms may be seen during the course of illness. ^{42 -44} While subtle personality changes, disinhibition and problems in executive functioning ⁴⁵ are frequently encountered in FTD, frank psychotic symptoms resembling schizophrenia are unusual. Loy et al ⁴⁶ presented the case of a 48-year-old woman with an 18-month history of bizarre and complex delusions on a background of social, behavioral, and cognitive decline over several years. The patient believed that she had become pregnant following a 2-week sexual relationship with an intruder in her house. The delusion persisted despite treatment with risperidone and, subsequently, with olanzapine. Neurological examination revealed frontal release signs, as well as increased jaw jerk and generalized brisk limb reflexes. Magnetic resonance imaging (MRI) demonstrated mild, bilateral atrophy of the frontal lobes. In terms of neuropsychological deficits, the patient had predominantly executive dysfunction. The signs and symptoms satisfied the clinical diagnostic criteria for FTD. ⁴⁷ When the patient died of pneumonia 4 months after admission to hospital, autopsy was performed. She was found to have FTD with ubiquitin-positive and TAR DNA-binding protein 43 (tDP-43)-positive pathology. Snowden et al ⁴⁸ have noted that psychosis in patients with FTD is strongly associated with a hexanucleotide repeat expansion in C9ORF72 gene. A 42-year-old woman with change in personality and with prominent delusion of pregnancy as an early feature of frontotemporal lobar degeneration with motor neurone disease was presented by Larner ⁴⁹ to illustrate the need to consider neurodegenerative disease as well as primary psychiatric disorder as the underlying cause of this striking symptom. A posthumous examination of the patient’s stored DNA was undertaken. The abnormal C9ORF72 hexanucleotide repeat expansion was detected, confirming the previous clinical diagnosis and establishing its genetic etiology. ⁵⁰ The presence of delusions in behavioral variant FTD (bvFTD) is intriguing because of the potential underlying mechanisms and the clinical similarities to schizophrenia. ^51,52 At times, this phenotypic overlap can mean that schizophrenia and FTD are 2 possibilities in the differential diagnosis of a psychotic presentation. ⁵³ Young persons presenting unusual behavior, emotional blunting, social withdrawal, declining psychosocial functioning, and executive dysfunction with or without frontotemporal changes in imaging are likely to receive a psychiatric diagnosis of schizophrenia. ⁵² Although schizophrenia and FTD have been typically described in terms of specific feature constellations characteristic for each disorder (eg, younger onset and more pronounced delusions as well as hallucinations in schizophrenia), it has also been shown that there is a great deal of clinical, neuroimaging, genetic, and pathological overlap, making it sometimes difficult to distinguish between these 2 conditions. ⁵⁴ Some reports ^55,56 illustrate how, in its early stages, FTD may be confused with schizophrenia. This overlapping phenotype is indicative of dysfunction of similar brain systems and pathways. ³⁹ Frontal, temporal and hippocampal atrophy, ⁵⁷ and regionally specific reductions in the anterior corpus callosum ^58,59 and anterior hippocampus ^60,61 have been described in MRI studies of both FTD and schizophrenia. There is not a shared convincing evidence in support of FTD rather than a psychosis involving the frontal lobe. Only with time, the progressive nature of FTD generally clarifies the diagnostic confusion, but it is clear that a subset of patients have an ambiguous presentation at the onset of symptoms. ⁵³ The presence of a relentlessly downhill course progressing to a profound global dementia and death rather than a moderately severe end state will in time suggests FTD rather than simple schizophrenia but may not be helpful diagnostically at the outset. ⁵⁵ Genetic findings could be an aid to the correct diagnosis, since current criteria lack sensitivity in the early stages of the disease, ⁴⁸ but potential links between the 2 disorders have also been demonstrated at molecular and genetic levels. According to Cooper and Ovsiew, ⁵³ although there is much clinical overlap, some features can help distinguish these disorders. A profound loss of empathy is a diagnostic feature of bvFTD not commonly seen in schizophrenia. Although overeating can be seen with antipsychotic treatment, hyperorality or compulsive eating—particularly a predilection for sweets—is indicative of bvFTD. Loy et al ⁴⁶ proposed some clinical features differentiating late-onset schizophrenia from FTD, for example, the presence of hallucinations (predominantly auditory hallucinations in late-onset schizophrenia, rarely present in FTD and when present they occur in nonauditory modalities) and antipsychotic response (good in late-onset schizophrenia, unknown in FTD). No study was performed that specifically sought to investigate the prevalence of a diagnosis of schizophrenia or other psychotic illness during the early stages of FTD. ⁵²

Treatment and Prognosis

There is no specific treatment for delusions of pregnancy. Patients with delusional pregnancy have been reported to be more hostile and treatment resistant compared with matched controls. ⁶² However, psychotropic drugs may play a key role along with the treatment of medical comorbidities. Antipsychotic medications (flufenazina, ¹⁹ haloperidol, ^19,21,28 trifluperazine, ²⁸ chlorpromazine, ²³ amisulpride, ³⁹ and risperidone ⁶³ ) were successfully used, but in other cases, delusion progressed despite neuroleptic treatment. ⁴⁶ Treatment of hyperprolactinemia must be conducted when faced with a delusion of pregnancy. When hyperprolactinemia is observed, a prolactin-sparing antipsychotic agent (such as aripiprazole) is the first choice. ³⁶ Soygür et al ⁶⁴ described the case of a young female with hypophyseal microadenoma and delusional pregnancy induced by hyperprolactinemia. She was successfully treated with the antipsychotic melperone, an atypical butyrophenone that produces a marked blockade of central dopamine D2 receptors inducing clinically relevant antipsychotic activity with rare extrapyramidal side effects and hyperprolactinemia. ⁶⁵ There is support for use of electroconvulsive therapy (ECT) in patients with psychotic depression. ²⁸ Bernardo et al ⁶⁶ described a patient with psychotic depression with delusion of pregnancy. After 6 weeks of treatment with haloperidol (up to 14 mg a day) and imipramine (up to 250 mg a day) without clinical improvement, the symptoms completely resolved with a course of 12 ECT. Cognitive psychotherapy can focus on cognitive processing of sensory experiences and can help patients to correct irrational beliefs and hypothesis. ²⁹ There has been one reported case of delusion which lasted for over 15 years. ³⁰ However, the prognosis is related to the cause that led to the delusional symptomatology. For example, the psychotic symptoms in FTD are persistent, and sometimes resistant to treatment. ⁶⁷

Conclusions

Delusion of pregnancy is a bizarre form of delusion due to organic, functional, or drug-induced causes. It is reported among DPS that consists of a sequence of delusions in the cycle of human procreation. Psychotic phenomena such as delusions may be an early feature in some cases of dementia. If delusion of pregnancy is an early presentation of FTD whose usual onset is in the presenium, it may be initially misdiagnosed as psychotic disorder.