Case Description
We present the case of a
55-year-old female patient with past history of a non-specified depressive
disorder with irregular psychiatric follow-up, as well as a history of alcohol
dependence. One month prior to the admission she underwent an orthopedic knee
surgery and decided to suddenly stop both the alcohol intake and the
psychiatric medication. The surgery was uneventful, and she was discharged
home. Until the presentation on emergency department the family reported subtle
behavioral changes, as repetitive actions and slight irritation. She was
brought to the ED after suddenly displayed erratic and frantic behavior,
claiming that one of her children was going to be assaulted, despite every
proof otherwise. On physical examination she was severely agitated and in acute
distress with paranoid speech. The remaining examination was unremarkable and a
cerebral computer tomography scan didn’t show any lesions. The laboratory
results reveal only a light anemia and leukocytosis with a negative C reactive
protein (Table 1).
With necessity of
beginning sedative medication an electrocardiogram (ECG) was performed
revealing a sinusal rhythm with a ST elevation on leads V2 and V3. A high
sensitivity I troponin becomes high elevated with no elevation on brain natriuretic
peptide (Table 1). A summary echocardiogram showed LV systolic dysfunction with
apical akinesia. She was admitted to the Cardiology Unit, where she stay
asymptomatic on cardiac aspect, with maximum troponin I-hs on the second day. A
coronary angiography performed that day didn’t show any significant coronary
disease but the left ventriculography reveals apical and mid-wall hypokinesis
suggestive of TS (Figure 1).
She started on acetylsalicylic acid 100mg/day, bisoprolol 2.5mg/day and lisinopril 2.5mg/day with good clinical evolution and resolution of the ECG abnormalities. An echocardiogram at day 9 showed recovery of LV systolic function and she was discharged from cardiology unit with assessment 1 month later for revaluation. Related to psychiatric disease, the patient continued to exhibit altered behavior and paranoid speech, experiencing primary persecutory delusions, delusional perceptions and delusional memories. She was then transferred to a Psychiatric inpatient service under treatment risperidone 2mg/day. During her stay at the Psychiatric unit, the patient did not tolerate risperidone’s side effects of sedation and motor retardation, so it was switched to aripiprazole with good response, with residual primary delusion until the end of the admission. The patient was discharged home with the diagnosis of schizoaffective disorder.
Table 1: Blood serum analytical results at the emergency department, on cardiology unit at day 2 and at discharge.
|
|
Emergency department |
Day 2 of admission |
Discharge |
Reference value |
Unit |
|
Hemoglobin |
11,2 |
|
10,7 |
12,0-16,0 |
g/dL |
|
Mean globular volume |
92,6 |
|
|
87-103 |
fL |
|
Leucocytes |
16,41 |
|
7,64 |
4,0-11,0 |
x109/L |
|
Neutrophiles |
85,1 |
|
|
53,8-69,8 |
% |
|
Lymphocytes |
9,1 |
|
|
22,9-36,6 |
% |
|
Platelets |
412 |
|
274 |
150-400 |
x109/L |
|
Urea |
19 |
|
21 |
10-50 |
mg/dL |
|
Creatinin |
0,89 |
|
0,69 |
0,51-0,95 |
mg/dL |
|
Sodium |
133 |
|
141 |
135-137 |
mEq/L |
|
Potassium |
3,5 |
|
4,5 |
3,5-5,1 |
mEq/L |
|
C reactive Protein |
4,2 |
|
|
<3,0 |
mg/L |
|
Creatine kinase |
115 |
|
|
10-149 |
U/L |
|
Myoglobin |
371 |
|
|
<146 |
ng/mL |
|
Troponin I - hs |
1285 |
8216 |
|
<16 |
ng/L |
|
Brain natriuretic peptide (BNP) |
18,3 |
|
|
<100 |
pg/mL |
Figure
1: a) Ventriculography of left ventricule in systole
with apical and mid-wall hypokinesis. b) Ventriculography in diastole.
Discussion
Takotsubo syndrome (TS)
is an acute and reversible wall motion abnormality of the left or right
ventricular myocardium. The condition was named after the Japanese octopus pots
(“takostubo”) that resembles the characteristic left ventricular end-systolic apical
ballooning shape that can be seen on the transthoracic echocardiogram or on the
ventriculography of these patients [1]. Typically occurs in post-menopausal
women and is usually elicited by an identifiable emotional or physical stress
in about two thirds of cases [1,2]. Although its etiology is still not
completely understood, the most widely reported hypothesis is a direct
catecholamine-mediated myocardial stunning, caused by high levels of serum
catecholamines. Other possible mechanisms include multivessel vasospasm,
estrogen induced and cardiovascular stress responses [1]. TS is usually
manifested by symptoms similar to those of an acute coronary syndrome (ACS),
such as acute substernal chest pain, nausea, syncope, dyspnea or palpitations.
On ECG we will find usually ST-segment elevation, T-wave inversion or QTc
prolongation, sometimes with temporal evolution as seen in ACS. The biomarkers
are usually elevated, with troponin values equally elevated compared to ACS
[2,3]. The diagnose in our patient was particularly challenging since she
didn’t report any symptoms. Although the presence of troponin elevation, ECG
and echocardiogram findings poses a suspicion on TS, the final differential
diagnoses was made by coronary angiographic and ventriculography. These shows
absence of either obstructive coronary artery disease or acute plaque rupture
and the observation of the typical left ventricle contraction pattern, on
ventriculography [3]. The recommended treatment is supportive with management
of complications. Prognosis in the acute phase is similar to that of ACS, with
approximately 30% of patients having serious cardiac complications but unlike
them, in TS a full recovery of the left ventricular function occurs in days to
several weeks [2,4].
In recent years, there
has been as increasing number of literature linking pre-existing or active
psychiatric disease and TS. Of note the majority pre-existing are related with
mood or anxiety disorders with few cases related with other psychiatric
illness, including schizophrenia-spectrum and other psychotic disorders [2,4].
Related to an “acute” presentation or exacerbation of psychiatric disorders, in
the International Takotsubo Registry only 10% of patients were diagnosed with
“acute psychiatric disorders”. Despite this, the proportion of TS clearly
triggered by acute psychiatric decompensation remains unknown [4]. The
pathology behind these association is largely speculative with the most
reported mechanism being an interaction between the brain and heart mediated by
autonomic nervous system activation and an increase blood catecholamine levels
[1,5]. Although some psychotropic medications have also been associated as
triggers on TS, most of them in the setting of relatively rapid uptitration or
in overdose [4], in the case that we report, this association is not a possible
mechanism since the patient stopped all the medication 1 month before. TS has
been described in context of acute alcohol withdrawal to, for the case
presented is not a possibility mechanism since she suspended all the consume 1
month before [5].
There are no particular
orientations in the literature towards the treatment of acute psychiatric
illness in the context of TS. This is particularly true in the case of
psychotic disorders, since it is such a rare phenomenon. Accordingly, the
treatment of our specific patient was oriented by general guidelines and
recommendations, taking into account her clinical picture and comorbidities.
Risperidone was initially chosen because of its anti-psychotic efficacy,
anxiolytic properties, long-term metabolic effects and low risk of QTc interval
prolongation, compared to other anti-psychotics [6].
Conclusion
Our case adds important
information to the rare interaction between psychosis and TS. Also, to the best
of our knowledge, this is the first reported case of TS as a manifestation of a
first episode psychosis in the context of schizoaffective disorder.
References
6.
Taylor
DM, Barnes TR, Young AH. The Maudsley Prescribing Guidelines insychiatry. John
Wiley & Sons. 2018.