New Journal and we have not received input yet 23–24 (2020) 100064

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Personalized Medicine in Psychiatry

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A challenging case of catatonia during pregnancy

Natalie Martinez-Sosa *, Joshua Delaney, Stephen McLeod-Bryant
Department of Psychiatry and Behavioral Sciences, University of Miami Miller School of Medicine, Miami, FL 33136, USA

A R T I C L E I N F O A B S T R A C T

Keywords: Co-occurring pregnancy and catatonia is a challenging combination to treat as the first line treatment for
ECT catatonia, benzodiazepines, have been shown to have negative effects on a fetus. ECT is another recommended
Catatonia treatment in this patient population but was not available on admission. The patient was treated with risperidone
Pregnancy
and lorazepam, which was deemed ineffective, and memantine was started. Shortly after, ECT became available
and in combination with memantine, catatonia was treated effectively.

1. Introduction
Multiple case reports have documented catatonia in the setting of
psychotic illness but there are few reports of catatonia during the pre­
natal period. Multiple etiologies can lead to the development of cata­
tonia, including medical, psychiatric, and neurologic, although most
frequently catatonia has been observed in patients with an underlying
mood disorder. First-line treatment involves the use of benzodiazepines;
electroconvulsive therapy (ECT) has been viewed as first-line in cases of
severe or malignant catatonia[1] and is thought to be the favored choice
during pregnancy due to the teratogenic risk of benzodiazepines. We
present the case of a patient with unknown underlying psychiatric
illness who presented catatonic during her first trimester of pregnancy.
2. Case report
A 21 year-old female originally presented to an outside hospital in
her first trimester of pregnancy at 11 weeks gestational age with a two-
month history of psychomotor retardation, increased speech latency,
and withdrawn behavior. The patient already had a guardian advocate
appointed by the time she arrived at our institution and permission to
obtain outside medical records was denied. The few documentation
available to us from her prior hospitalization indicated the patient had a
questionable history of schizophrenia and was started on risperidone 1
mg in the morning and 3 mg at bedtime. On admission to our hospital,
she was awake and alert, though intermittently mute with increased
speech latency, prominent psychomotor retardation, flat affect, and
urinary incontinence. On admission, medical workup included complete
blood count that revealed an elevated white blood count of 14 and
elevated neutrophils (81.4%) consistent with an infectious process while
complete metabolic profile and lipid profile were within normal limits.
Urinalysis revealed the presence of leukocyte esterase, ketones, and
bacteria, consistent with a urinary tract infection and was subsequently
started on nitrofurantoin. She was found to have syphilis and subse­
quently treated with penicillin G. The patient was also started on pre­
natal vitamins and folic acid 3 mg daily. CT brain without contrast and
urine toxicology were both negative and physical examination per­
formed in the emergency department and in the unit were both unre­
markable. Patient’s boyfriend reported the patient had been doing well
and was employed before her admission to the prior hospital. He denied
any psychiatric history but noted two months of her having staring ep­
isodes, poor sleep, not bathing herself, isolating, and only giving short
answers. He also endorsed a history of substance abuse including
cannabis.
On the unit, the patient was found to be catatonic; scoring 18 on the
Bush-Francis Catatonia Scale (BFCS), significant for mutism, staring,
posturing, immobility/stupor, mild rigidity, negativism, waxy flexibility
and ambitendency – where she seemed “stuck” in an indecisive move­
ment or showed hesitancy when doing something. Vitals signs remained
stable during hospitalization and obstetrics was consulted for prenatal
care. Given that ECT was not immediately available and self-care was
poor, lorazepam 1 mg daily was added to risperidone and increased to 3
times a day with little to no improvement over the duration of approx­
imately 10 days. Risperidone was discontinued due to unclear history of
psychosis, lack of improvement on medication, and increased risk of
malignant catatonia. Due to risk of teratogenicity lorazepam was slowly

* Corresponding author at: Department of Psychiatry and Behavioral Sciences, University of Miami Miller School of Medicine, 1695 NW 9th Ave Miami, FL 33136,
USA.
E-mail address: natalie.martinezsos@jhsmiami.org (N. Martinez-Sosa).

https://doi.org/10.1016/j.pmip.2020.100064

Available online 13 November 2020

2468-1717/© 2020 Elsevier Inc. All rights reserved.
N. Martinez-Sosa et al.
tapered down and memantine was started at 5 mg daily. Three days after
starting memantine the patient had asked to go to the bathroom for the
first time since being admitted and continued to be continent throughout
the rest of her stay. Memantine was titrated to 10 mg in the morning and
5 mg at night while waiting to see if ECT was an option at another
hospital. Staff noticed slight improvement in the patient’s condition.
After approval from the mental health court, due to her incapacity,
contractual arrangements were made with a local hospital that could
provide ECT, and the patient received 7 sessions of bilateral ECT with
good response, with significant improvement seen after the fourth ses­
sion with a BFCS of 7. During ECT her medication regimen consisted of
memantine 10 mg twice daily and lorazepam 1 mg twice daily. Once the
patient’s symptoms began to improve more history was obtained from
the patient. She stated her symptoms began as anxiety leading to
depression, prior to hospitalization. She identified psychosocial stressors
exacerbating her anxiety and depression, including her work and having
to care for her boyfriend’s children. She noted becoming withdrawn, not
eating, isolating self from family, and spending increasing amounts of
time in bed. She denied any history of psychotic symptoms or symptoms
concerning for prior episodes of mania or hypomania. Patient did report
a history of physical abuse from her mother, which led to her being
adopted at a young age but denied any symptoms concerning for post-
traumatic stress disorder. She was also unaware of family history of
mood or psychotic disorders. She endorsed a history of cannabis use,
starting at the age of 18 and using up to 3.5 g daily leading up to her
admission to the outside hospital which was approximately one month
before being transferred to our hospital.
Upon discharge, lorazepam and memantine were discontinued and
the patient was discharged with outpatient psychiatric and obstetrics
follow-up. Months later the patient was evaluated by the CL Psychiatry
team when she presented in labor with premature rupture of membranes
(PROM). She denied any symptoms of depression, psychosis, mania, or
anxiety. She had a normal spontaneous delivery at 31 weeks and was
discharged on post-partum day 2. Patient declined any psychotropics at
that time but was aware of the possibility of return of her symptoms. The
baby girl stayed in the hospital for 50 days. She was found to have hy­
aline membrane disease, apnea of prematurity, retinopathy of prema­
turity and anemia of prematurity. On discharge, apnea of prematurity
resolved, retinopathy of prematurity showed no vessels to zone III and
the anemia of prematurity was resolving after being treated with Epogen
and iron supplementation.
3. Literature review
Catatonia has been described as a syndrome with motor signs typi­
cally manifested as mutism, negativism, posturing, rigidity, staring, lack
of response to pain, repetitive movements, stupor, as well as purposeless
agitation[2]. The pathophysiology of catatonia includes psychiatric,
neurologic, and medical causes, which are thought to cause abnormal­
ities in neurotransmitters including dopamine, GABA, and glutamate[3].
Catatonia is frequently undetected in the clinical setting and rates vary
according to study design, however incidence in patients hospitalized
with acute psychotic episodes is within the 7–17% range and 13–31% in
mood disorders[4]. Catatonia occurs most commonly with mood dis­
orders and more than half of patients with catatonia have an underlying
bipolar affective disorder[1]. The diagnosis is made based on clinical
signs although there are scales that can be useful to screen for catatonia
including the Bush-Francis Catatonia Rating Scale.
The literature on catatonia during pregnancy is minimal. Nonethe­
less, it is known that pregnancy and post-partum are periods of increased
vulnerability in patients with psychiatric illness[5]. Worsening psy­
chopathology in the prenatal period can lead to neglect in prenatal care,
malnutrition, risk of preterm delivery and low weight at birth[6]. More
specifically, prolonged catatonia can lead to medical complications
including pressure ulcers and increased risk of thromboembolism;
nutritional deficiencies and dehydration are also common due to poor
2
Personalized Medicine in Psychiatry 23–24 (2020) 100064
dietary intake[7]. It is of utmost importance that the risks and benefits of
treatment, either with ECT or psychotropics, be weighed against the risk
of untreated psychiatric illness when making decisions for management
in this patient population.
Catatonia can resolve completely when properly treated. Benzodi­
azepines, particularly lorazepam, and ECT are the most frequently rec­
ommended treatments. Combination treatment with benzodiazepines
and ECT might be clinically effective as they both result in increased
seizure threshold and decreased cortical excitability[1]. However, ECT
is the most effective as it has been demonstrated to induce remission
when benzodiazepines have failed and are effective for mood or psy­
chotic disorders associated with catatonia[4]. However, in some cases,
ECT is not a possibility. This has led to other interventions including the
use of NMDA antagonists, zolpidem, antiepileptics, and atypical anti­
psychotics[8].
NMDA antagonists are a class of medications that have evidence of
success in treating catatonia; namely amantadine and memantine. The
way these medications treat catatonia is still unknown. In rats, aman­
tadine has shown to increase embryonal death at higher than maximum
doses while shown to increase visceral and skeletal malformations at
maximum doses[9]. There are few reports of teratogenicity in humans
using amantadine. Animal studies using memantine showed association
with maternal toxicity, decreased vertebral ossification, and lower pup
weights when administered at a much higher level than the maximum
recommended human dose (MHRD), but when given three times above
MHRD there was no effect seen. There was no teratogenicity with more
than maximum dose and there have been no reports of teratogenicity in
humans[10]. The data and safety of using these medications in preg­
nancy is scarce.
Catatonia in pregnancy is seldom described in literature although
case reports have shown effective treatment with benzodiazepines and
ECT. However, benzodiazepines may be limited in their use during
pregnancy due to increased risk of oral cleft, more commonly seen with
diazepam, and of neonatal abstinence syndrome. Benzodiazepines have
also been associated with increased risk of spontaneous abortion in early
pregnancy, with the risk being greater at increasing daily doses[11].
Meanwhile, ECT has been reported a safe and effective treatment during
pregnancy due to decreased fetal exposure to teratogenic medications
[1]. Multiple reviews have come to the conclusion that ECT is a safe
alternative during pregnancy and there are two case reports of ECT
working in a pregnant patient with catatonia[12–14], however the
volume of data regarding its safety during pregnancy is still limited. A
careful evaluation of risk and benefits should still be taken into
consideration as ECT has been associated with placental abruption,
uterine contractions, and reduced fetal heart rate[15].
4. Discussion
One of the challenges presented in this case is the lack of a known
psychiatric or non-psychiatric history making the etiology of her cata­
tonic state either psychotic in nature or due to an underlying mood
disorder. We know the patient was diagnosed with syphilis at the time
which could have manifested as neurosyphilis leading to psychosis.
However, the patient received treatment with penicillin but no lumbar
puncture was done to completely rule out this diagnosis. Furthermore,
her psychiatric symptoms persisted despite adequate treatment. The
patient had been prescribed psychotropics at the previous facility, which
could indicate a psychotic etiology or an affective disorder with wors­
ening catatonia in the setting of prolonged exposure to psychotropics.
ECT was not an option at the beginning of treatment making psycho­
tropics the only possibility to treat the patient. We discontinued the
risperidone that the patient was initially treated with due to poor
response, increased risk of complications and the possibility of it wors­
ening her catatonia. Lorazepam was started, as it is indicated in the
treatment of catatonia, however due to poor response and instead of
continuing to increase it, other options were explored. Antiepileptics
N. Martinez-Sosa et al.
have been used off-label to treat catatonia, however some agents in the
class, particularly carbamazepine and valproic acid, have been associ­
ated with neural tube defects in the developing fetus. Zolpidem has
shown to have adverse effects on the development of offspring above 10
mg and treatment of catatonia usually requires greater dosing of 10 mg
[16,17]. Literature review has shown memantine, when compared with
other off-label agents, to have a side effect profile that one might deem
less damaging, and has had reported success in catatonia. After initiating
treatment with memantine the patient began to show slight improve­
ment, however ECT also became an option at that time. She received a
total of 7 sessions of bilateral ECT with the most robust response coming
after the fourth session.
The patient experienced PROM at approximately 31 weeks. It is
unclear what may have caused this as the patient had been on different
medications, undergone ECT and was experiencing symptoms that have
been shown to be responsible for premature delivery. In regards to the
medications the patient was exposed to, a case control study has shown
benzodiazepines to be linked to premature birth but not low birth
weight[18]. A review of risperidone use during pregnancy states that in
utero exposure does not appear to increase the risk of spontaneous
abortion or fetal teratogenic risk when compared to the general popu­
lation, however, it did not elaborate as to risk of preterm labor[19].
Other cases have reported premature labor while ongoing ECT treatment
thus stopping ECT[15], however, our patient had no problems during
her ECT course and PROM happened approximately 11 weeks after her
last ECT so it is unlikely ECT was the culprit. Nevertheless, it has been
shown that pregnant women with depression are more likely to have a
preterm birth compared to non depressed women [20], however, the
cause of the PROM in our patient is unknown and seems unlikely to be
related to her previous mental illness or its treatment.
This case report adds to the literature on treatment of catatonia
during pregnancy showing resolution of symptoms after multiple ses­
sions of ECT. It also illustrates the need for further studies in the field
and the development of guidelines to help aid management in this
population.
Declaration of Competing Interest
The authors declare that they have no known competing financial
interests or personal relationships that could have appeared to influence
the work reported in this paper.
Personalized Medicine in Psychiatry 23–24 (2020) 100064
Appendix A. Supplementary data
Supplementary data associated with this article can be found, in the
online version, at https://doi.org/10.1016/j.pmip.2020.100064.
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