Abstract
Chief Complaint and Presenting Problem
A was a 9-year-old girl with a history of oppositional defiant disorder (ODD) and attention-deficit/hyperactivity disorder (ADHD) who was initially referred to the emergency department (ED) for increasing agitation and complaints of being pregnant.
History of Present Illness
On initial evaluation in the ED, mother reported that A. had been stating she was pregnant for the past 4 days. There had been no psychosocial precipitants or relevant stressors. Despite reassurance that she was unable to become pregnant, A. had continued to believe she was pregnant. As she became more and more convinced of the pregnancy, mother reported that A. became aggressive when her belief was challenged. By day 4, A. reportedly experienced some abdominal pain, which she attributed to the babies' (twins) impending delivery, and requested that mother take her to the hospital. Mother reported that she obliged because she was concerned with A.'s delusional thinking.
In the ED, A's abdominal pain was evaluated. Vital signs were reported to be within normal limits. General laboratories were completed, and complete blood count, blood chemistry, liver function tests, and lipase all returned within normal limits. Pregnancy test was negative. On examination, A.'s abdomen was distended but nonacute, soft, and nontender. At that time it was felt that no further medical workup for the abdominal pain was indicated, and A. was admitted to the inpatient child and adolescent psychiatry unit due to concerns for safety.
On admission, A.'s medications included citalopram 30 mg daily and risperidone 1 mg twice daily. A. had been on a stable dose of risperidone for 6 months, but the citalopram had been increased from 10 to 30 mg over a 2-week period before ED referral. In addition to the new onset delusional thinking, mother reported increased restlessness, decreased need for sleep, and agitation with the recent dose change.
Psychiatric History
A. had been diagnosed with ODD at age 3 years. At age 5 she was diagnosed with ADHD, and had received individual therapy as treatment. Per records, A. was admitted to a day treatment program during Kindergarten and subsequently for 6 weeks to a residential treatment program. She began medication treatment for ADHD at age 7. At age 7, A. was readmitted to a residential treatment center (RTC) for 8 months, and subsequently for an additional 3 months.
Developmental History
Records indicate the pregnancy was complicated by maternal use of cocaine. There were no known complications during birth. Birth weight was unknown; the baby went home with mother without an extended hospital stay. Motor milestones were reported as met on time. Language acquisition was reportedly delayed.
Educational History
A. reportedly received early intervention service, but she did not receive a special education preschool program. A. was admitted to a day treatment program during kindergarten. She was reported to have switched schools frequently throughout elementary school.
A. had reportedly completed second grade at the time of evaluation. Per her Individualized Education Program, an occupational therapy evaluation indicated intact visual and fine motor skills. The teacher report indicated moderate sensory-seeking behaviors within the school setting.
Cognitive skills were reported as significantly below average. Further assessment using a nonverbal test of intellectual functioning indicated nonverbal intelligence quotient score in the low average range. Achievement for reading was in the very low range; achievement for mathematics and written language was in the low range.
Social History
A. lived with mother and a younger brother, age 2 years. There was no known history of sexual abuse. There was one prior report to child protective services due to repeated visits by A. to the school nurse for various somatic complaints. Upon investigation, there was no maltreatment substantiated. Father was reportedly not involved in the A.'s life and was currently homeless.
Family History
Father had reportedly been diagnosed with schizoaffective disorder. Maternal grandmother had been diagnosed with anxiety.
Medical History
A. had a history of chronic constipation and obesity. She had no hospitalizations or surgeries. She reportedly had experienced no major childhood illnesses, and had received all of her vaccinations on time.
Mental Status Examination
On initial examination, A. was agitated and restless. She appeared disheveled with unbrushed hair, pacing around the room. She refused to engage in the interview and responded shouting “I don't know” to most questions. There was no eye contact throughout the interview. She was ruminative on the delusion that she was pregnant. Speech was disorganized.
A. appeared to be responding to internal stimuli and aimlessly roamed the room, setting up “baby beds” with blankets. She appeared to calm down when discussing her babies, and she reported hallucinations of hearing her babies crying and feeling them moving in her stomach. A. denied suicidal or homicidal ideation. There were no other pertinent findings on mental status examination.
Hospital Course
During hospitalization, a complete medical workup for altered mental status was performed. A.'s weight was 48.8 kg and body mass index was 24.4 kg/m2. Routine electroencephalogram was notable for no epileptiform activity. Magnetic resonance imaging of brain and brain stem with and without contrast was normal. Abdominal radiograph was consistent with moderate stool burden; abdominal ultrasonography was completed without concern for masses. Lumbar puncture appeared bloody with 4000 red blood cells, 50 segmented neutrophils, and 45 protein. Glucose was normal. Cerebrospinal fluid (CSF) infectious laboratories including herpes simplex virus, varicella-zoster virus, Mycoplasma pneumoniae, enterovirus, cytomegalovirus, West Nile, and venereal disease research laboratory were negative. CSF encephalopathy laboratories including serum ENC2 and a limbic encephalitis panel were also negative.
In the hospital, A.'s citalopram was discontinued. Her risperidone was increased to 1.75 mg. twice daily. A. exhibited new, bizarre, and disorganized behaviors, such as grabbing fecal matter in the toilet and inserting her hand into her vagina to remove the baby. A. became more aggressive, screaming, and attempted to hit staff. Consequently, over a 1.5 week period, risperidone was tapered and discontinued and chlorpromazine was started at 50 mg and gradually increased to 150 mg twice daily. A.'s agitation improved, and her delusions started to attenuate.
Just shortly after risperidone was discontinued, prolactin was elevated at 45.4 ng/mL (normal range for nonpregnant females is 2.8–29.2 ng/mL). After remaining on chlorpromazine, A.'s prolactin level decreased to 27.6 ng/mL at 2 months follow-up, and then to 7.8 ng/mL 2 weeks later. The delusions resolved once prolactin levels normalized.
Brief Formulation
In summary, A. was a 9-year-old prepubertal girl who met diagnostic criteria for a brief psychotic episode. Initially, her behavior was disorganized and bizarre, and thought content was notable for a delusion of being pregnant with twins. She was observed to be responding to internal stimuli and reported auditory and tactile hallucinations related to her delusion. In retrospect, A. had a long history of irritability and aggression since age 3 years, but no history of psychosis. There was no history of trauma. There had been some discrepancy in intellectual function in the past, but testing during hospitalization placed her in the low average range.
From a biological perspective, there was a paternal family history of schizoaffective disorder, increasing her risk for the development of a psychotic disorder. Medical workup for A.'s altered mental status revealed no other biological causes of her psychotic symptoms. From a psychosocial perspective, it is possible that A.'s residential center admissions and mother's absence and inconsistent attachment during early childhood may have rendered A. unusually competitive with her 2-year-old brother for mother's affection. This dynamic may have contributed to overdetermination of her desire to have her own baby.
Interestingly, A.'s behavior and agitation worsened with increases in risperidone, and improved after discontinuation of risperidone and initiation of chlorpromazine. Although on risperidone A. was noted to have an elevated prolactin level, as expected, the level started to fall after discontinuation of risperidone. As her prolactin level returned to normal, her delusions resolved.
Multi-Axial Diagnoses
Axis I: Brief psychotic disorder
Rule out bipolar affective disorder with psychotic features
Axis II: Deferred
Axis III: Constipation
Obesity
Hyperprolactinemia secondary to risperidone
Axis IV: Level of psychosocial stressors: moderate, multiple hospitalizations
Axis V: Current global assessment of functioning score: 10
Discussion
This is a very interesting clinical presentation and, to our knowledge, the first case of hyperprolactinemia-induced delusional pregnancy in a prepubescent girl. Although it is possible the increase in citalopram precipitated A.'s episode of mania as exhibited by her restlessness, agitation, and psychosis, these symptoms did not respond to discontinuation of citalopram and increase in risperidone as would be expected. There are a handful of cases of delusions of pregnancy in children reported in the literature, which cite psychosocial mechanisms and hyperprolactinemia as possible causes (Ali et al. 2003). Although literature reports chlorpromazine as a potential cause for hyperprolactinemia inducing delusions of pregnancy, this case demonstrates reduction in prolactin levels and resolution of psychotic symptoms with chlorpromazine.
Risperidone is known to increase prolactin through D2 antagonism. Selective serotonin reuptake inhibitors have also been shown to elevate prolactin through serotonergic effects, although reports have been mixed (Torre and Falorni 2007). It is plausible that the addition of citalopram to risperidone synergistically induced prolactin secretion in this individual.
Chlorpromazine has been reported to elevate prolactin through the tuberoinfundibular pathway through a decrease of prolactin-inhibiting factor. Reports of females with already elevated baseline prolactin have demonstrated no further increase in prolactin after chlorpromazine administration (Archer et al. 1977).
This case illustrates a child whose prolactin levels decreased with the addition of chlorpromazine. It is plausible that since A.'s prolactin was already elevated, initiating chlorpromazine may have caused a negative feedback loop, halting further secretion. It is also plausible in this case that chlorpromazine had less affinity for these D2 receptors on lactotrophs than risperidone or citalopram.
In conclusion, this case illustrates the potential utility of chlorpromazine treatment in prepubertal children who have delusions of pregnancy attributed to hyperprolactinemia.
Footnotes
Acknowledgment
We acknowledge the editorial assistance from Maria Cruz, BA.
Disclosures
B.J.C. is on the scientific advisory board of Abide Therapeutics and Teva/Nuvelution, received honoraria from the American Academy of Child and Adolescent Psychiatry, and received research support from Neurocrine Biosciences and NIM-H/UCSF. She is cochair of the medical advisory board of the Tourette Association of America (TAA), and on the speakers' bureau for the TAA-CDC partnership. The other authors have no other disclosures.