Hope or desperation? A pseudocyesis in advanced maternal age in a woman with recurrent miscarriages: a case report and literature review

¹Unit of Obstetrics and Gynaecology, Department of Primary Health Care Sciences, Faculty of Medicine and Health Sciences, University of Zimbabwe, ²Sally Mugabe Hospital, Harare, Zimbabwe

^&Corresponding author
Grant Murewanhema, Unit of Obstetrics and Gynaecology, Department of Primary Health Care Sciences, Faculty of Medicine and Health Sciences, University of Zimbabwe

Introduction    

This case has been reported according to the CARE guidelines [1]. A pseudocyesis refers to an impression by a woman that she is carrying a foetus, with associated pregnancy-related changes, when in fact she is not pregnant. A vexing psychological phenomenon, where a woman can exhibit full-blown pregnancy symptoms, and included in the Diagnostic and Statistical Manual for Mental Disorders Fifth Edition (DSM-5), the pathophysiology of pseudocyesis remains to be fully elucidated [2]. These false pregnancies were described as early as the seventeenth century [3]. They can occur in any racial, ethnic or socioeconomic group, though evidence suggests that they occur more commonly in societies where the cornerstone of marriage is childbearing [3]. Pre-menarchal and postmenopausal cases exist [4], though the 20 to 39 years age group is the most affected, and the exact prevalence is not clearly documented [2,3]. We report the case of a 44-year-old who presented to our unit with a strong conviction that she had an advanced pregnancy and was subsequently diagnosed to have a pseudocyesis.

Patient and observation     

History: a 44-year-old woman, in her tenth pregnancy, with a history of nine first trimester miscarriages, was admitted into our antenatal ward with a diagnosis of pre-eclampsia at an estimated gestational age of thirty-six completed weeks. She had booked her pregnancy late in the third trimester, and had gone to the clinic for a routine antenatal visit when she was noted to have an elevated blood pressure (BP) of 162/102 mmHg with significant proteinuria on urinalysis. She was referred to our hospital, a tertiary teaching institution in Harare, Zimbabwe for further management. A repeat blood pressure at the hospital was still elevated at 160/111 mmHg, despite an oral dose of 20mg of slow release nifedipine that had been administered at the local clinic. She was otherwise not complaining of any other symptoms, and she reported perceiving "normal" fetal movements from approximately 20 weeks of completed gestation.

Despite the several recurrent miscarriages, the patient had never been investigated and did not have a pre-conception visit. No pregnancy test was done when she missed her menses, and she was unable to book early due to financial constraints. No ultrasound scan was available. Antenatal HIV test was negative, but no other tests had been at the clinic where she had booked. She experienced early pregnancy symptoms, including nausea and vomiting, which stopped at the time when she had quickening, around 18-20 weeks. She was an otherwise well patient with no chronic illnesses, and had never undergone surgery. No evacuation of the uterus was done in any of her previous pregnancies. She had menarche at around 16 years of age, and married around 19 years of age when she had her first pregnancy. She was currently in her third marriage, in a polygamous marriage as her first two marriages had broken down because of the recurrent miscarriages. She was excited that this pregnancy had proceeded into the third trimester and she was looking forward to the birth of her baby. Her highest level of education was primary school, and she was a housewife, staying with husband and family in a high-density suburb. They were of poor socioeconomic circumstances, and she had to cope with very little support from the husband. They belonged to an apostolic sect.

Physical examination and evaluation: on admission, she was noted to have an elevated blood pressure but otherwise well, with a distended abdomen, in keeping with pregnancy. Initial results from her laboratory work up revealed normal liver enzymes, with alanine aminotransferase of 32 iU/l and aspartate aminotransferase of 22 iU/l. She had normal urea of 3.6 mmol/l and a creatinine of 72 μmol/l. However, she had a thrombocytopenia of 88/μL, with a normal haemoglobin of 13g/dL. An ultrasound of the pregnancy was ordered but could not be done on the day of admission. She was admitted for evaluation by the registrars and consultants. Seeing her the following day we noted a well patient with no abnormalities on physical examination. The abdomen was soft on palpation, and there was no palpable uterus above the symphysis pubis. An urgent USS showed a normal sized, non-gravid uterus.

Diagnosis and management: diagnoses of pseudocyesis and chronic hypertension were made, and patient was informed of the results. She cried with disappointment, and was appropriately counselled. She was referred to the internal medicine specialists for management of the hypertension and investigations for the thrombocytopenia, and to the psychiatrists for evaluation and psychotherapy. Before referral, we explored further her future fertility desires in view of very advanced maternal age and bad obstetric history. As she was still dealing with the current situation, we agreed that we would need to see her in the future in our outpatients clinic to investigate her fully and discuss her childbearing options.

Patient perspective: the patient was disappointed when she was informed that she was not pregnant. However, she was grateful that this has been discovered, and was hopeful that she would get appropriate help to navigate through.

Patients consent: written informed consent was obtained from the patient prior to discharge from the hospital.

Ethics statement: the local institutional review bodies, the Joint Research Ethics Committee of the University of Zimbabwe and Parirenyatwa Hospital and the Medical Research Council of Zimbabwe, require no permission to publish case reports.

Discussion     

We have presented the case of a woman who was desperately in need of a successful conception, and presented "near-term" with what she perceived to be a pregnancy. Subsequent clinical examination revealed that she was not pregnant, leading to the diagnosis of a false pregnancy, also known as a phantom pregnancy, pseudopregnancy or pseudocyesis. The presentation of this patient closely mimics what has been described in the literature [2]. The duration of a false pregnancy can range from anything from a few weeks, to nine months or more. The symptom complex ranges from early pregnancy symptoms such as nausea, vomiting, amenorrhoea, breast tenderness, to symptoms of established pregnancy such as quickening, abdominal distension and galactorrhoea [2]. The etiopathogenesis behind false pregnancies remains incompletely elucidated, but are postulated to involve a complex interplay of psychosocial, physiological and endocrinological factors [5,6].

Though the condition is listed in the DSM-5 for mental health disorders, according to Campos et al. the presentation of a pseudocyesis typically occurs out of mental health settings [2]. Somatic manifestations of pregnancy are precipitated by severe distress related to childbearing. We have presented a woman who had nine recurrent miscarriages, and two prior marriages, which failed due to failure to bear children. Her story represents that of women in African settings, where remarried women exhibit very strong desires for reproduction [7]. The primary goal for marriage in such cultures is for procreation and more so in a polygamous relationship where competition with other wives might result in her barrenness being a subject of ridicule. Her being in a polygamous union could have worsened her psychosocial distress, especially with the realisation of advanced maternal age.

Pseudocyesis must be differentiated from the delusions of pregnancy, which in the DSM-5 are categorised under the schizophrenic spectrum of psychiatric disorders [2]. Patients with delusions of pregnancy do not exhibit symptoms of pregnancy despite a grounded belief of an existing pregnancy, in contradistinction to those with pseudocyesis, who exhibit florid pregnancy symptoms. In a systematic review of cases of delusion of pregnancy by Bera et al. 35.7% of the patients had a diagnosis of schizophrenia, 16.7% were due to bipolar affective disorders, and 9.5% had a diagnosis of major depression [8]. The DSM-5 categorises pseudocyesis as a specified somatic symptom and related disorder, and defines it as a false belief of being pregnant, associated with objective signs and reported symptoms of pregnancy [9]. These may include abdominal enlargement, reduced menstrual flow, amenorrhoea, subjective sensation of fetal movements, nausea, breast engorgement and secretions, and labour pains at the expected time of delivery [2]. The patient we presented had symptoms closely resembling the pregnancy calendar, including quickening at 18-20 weeks, and represents the typical patient described in the DSM-5. The rarity of such patients has precluded observational studies with good sample sizes; hence, most are described in case reports. There may even be publication bias as some authors may consider them irrelevant.

Tarin et al. carried out a literature review to attempt to describe the endocrinology and physiology of pseudocyesis [5]. They noted endocrine similarities between pseudocyesis, polycystic ovarian syndrome (PCOS) and major depressive disorder (MDD) after excluding patients with delusions of pregnancy, though the traits were more akin to PCOS than MDD. Their data corroborated the notion of increased sympathetic nervous outflow activity, dysfunction of central nervous system catecholaminergic pathways involved in the regulation of hormone secretion from the adenohypophysis; and decreased steroid feedback inhibition of gonadotrophin-releasing hormone. These neuroendocrine/endocrine disorders may cause some of the symptoms highlighted above. However, these pathways do not explain the whole symptom spectrum in pseudocyesis, and the science is still to be fully elucidated [4].

Several case reports of false pregnancy have been published. However, some of the case reports describe cases that fit more into the spectrum of delusion of pregnancy as pseudocyesis. For instance, Talukder described two cases, both of which depict patients with underlying psychiatric problems and on treatment [10]. Their first case was of a woman who was convinced that she was seven months pregnant despite normal menstrual flows and was on antipsychotics. Their second case was of a woman who developed a depressive disorder on a background of subfertility, was put on antidepressants then went on to believe that she had fallen pregnant, with her symptoms resolving after psychotherapy and increased dose of antidepressants. However, Sultana et al. described a case of a true pseudocyesis, comparable to our case [11]. They presented a 37-year-old woman with a 21-year history of subfertility who presented with a history of 8 months amenorrhoea, and had experienced early pregnancy symptoms, but on subsequent examination her uterus was noted to be empty. Dashti-Kalantar et al reported the case of a patient who was rushed to theatre for an emergency caesarean section for antepartum haemorrhage and fetal distress at 28 completed weeks of gestation and was noted to have an empty uterus [12].

Our case, and those described in the literature, such as the one above where a patient had to undergo an emergency caesarean section whilst not pregnant [12], describe the prevalent deficiencies in the medical care for such patients, who book their "pregnancies" late. They have no proper antenatal care, where physical obstetric examinations and ultrasound scan would lead to early detection of their pregnancies. Medical models may not fully elucidate the underlying complex psychosocial contexts including the late presentations, therefore biopsychosocial models may be more appropriate. Appropriate counselling and psychotherapy are key for treating patients with pseudocyesis. However, some patients may require pharmacological interventions. Immediate involvement of psychiatrists is critical, to avoid further psychological complications. Our patient was extremely disappointed upon discovering that she was not pregnant, and we immediately involved psychiatrists, and also physicians to help manage her blood pressure. Multi-disciplinary approaches are key for the success of managing such complex patients.

Conclusion     

Pseudocyesis is a rare condition that may present to the unsuspecting clinicians. It must be suspected in patients with a longstanding history of subfertility who present with florid symptoms, especially without any assisted reproductive interventions. Counselling and psychotherapy are the cornerstones of treatment, though pharmacological treatments may be needed for some patients.

Competing interests     

The authors declare no competing interests.

Authors' contributions     

GM managed the patient and drafted the primary manuscript. AZ provided technical expertise in patient management and case writing, and critically revised the manuscript. MIN and MGM critically revised the manuscript. All authors read, finalised and agreed to publish the manuscript.

References