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Case report

Journal of the Intensive Care Society


2018, Vol. 19(4) 351–353
! The Intensive Care Society 2018
Leptospirosis presenting as severe Article reuse guidelines:
sagepub.com/
cardiogenic shock: A case report journals-permissions
DOI: 10.1177/1751143718754993
journals.sagepub.com/home/jics

E Forbat, MJ Rouhani, C Pavitt, S Patel, R Handslip and S Ledot

Abstract
Background: Leptospirosis is a rare infectious illness caused by the Spirochaete Leptospira. It has a wide-varying spectrum
of presentation. We present a rare case of severe cardiogenic shock secondary to leptospirosis, in the absence of its
common clinical features.
Case presentation: A 36-year-old woman presented to our unit with severe cardiogenic shock and subsequent multi-organ
failure. Her clinical course was characterised by ongoing pyrexia of unknown origin with concurrent cardiac failure.
She was initially managed with broad-spectrum antibiotics and inotropes. Percutaneous cardiac biopsy excluded major
causes of myocarditis. On day 21 after presentation, she was found to be IgM-positive for leptospirosis.
Conclusions: This is a rare case of severe cardiogenic shock secondary to leptospirosis infection. The case also highlights
the importance of obtaining a thorough social history when assessing a patient with an unusual presentation, as clues can
often be missed.

Keywords
Leptospirosis, cardiogenic, cardiac, shock

variable figures in neurological failure, although


Background neurological involvement is thought to be a predictor
Leptospirosis is an infectious illness spread via of mortality.1
rodents or mammals including dogs, monkeys and
wild boars. Spread can be via both direct and indirect
Case presentation
contact, and the disease is caused by the Spirochaetes
‘‘Leptospira’’ with a total of 14 possible pathogenic A 36-year-old woman was referred and then retrieved
species.1 The infection is most evident within tropical from her local general hospital due to new onset
regions such as Sri Lanka, with an annual incidence of severe biventricular failure. The patient presented
14/100,000 cases,2 and has low incidence in the UK; with a one-day history of upper abdominal pain and
a study in 2012 found 301 cases in the preceding five vomiting, following recent treatment for community-
years in the UK, a significant proportion of which acquired pneumonia. Her co-morbidities included
were acquired abroad.3 This has been attributed morbid obesity (body mass index [BMI] 53), diabetes
to the wide spectrum of disease severity and presen- mellitus type 2, asthma, hypothyroidism, chronic
tation, rendering it often underreported and under- back pain, depression, anxiety and previous alcohol
diagnosed. Risk factors, aside from endemic areas, excess and benzodiazepine dependence. She was also
include contact with urine of the vector, recreational an ex-smoker.
or occupational activity involving water and poor Abdominal computed tomography (CT) showed
hygiene. The onset of symptoms is quick and typically no features of intra-abdominal sepsis, and
occurs after an incubation period of five days to two
weeks.4The disease can present as either a mild, self-
limiting illness or be fulminant, with multi-organ Adult Intensive Care Unit, Royal Brompton Hospital, London, UK
failure most commonly neurological, renal and
Corresponding author:
respiratory. The mortality is variable upon the MJ Rouhani, Adult Intensive Care Unit, Royal Brompton Hospital,
organ dysfunction, with mortality rates of 30–60% Sydney Street, London SW3 6NP, UK.
in respiratory failure, 26–36% in renal failure and Email: maralrouhani@gmail.com
352 Journal of the Intensive Care Society 19(4)

subsequently her condition deteriorated over the next days. Repeat TOE suggested biventricular function
24 h with an acute systemic inflammatory response had recovered to a limited degree. From screening
syndrome, presumed to be due to an infectious pro- blood tests and further investigation into the cause
cess, metabolic acidosis (pH 7.27 (normal range [NR] of acute heart failure, the patient was found to
7.35–7.45), pO2 7.71 kPa (NR 10–14 kPa), pCO2 have biochemical evidence of hypothyroidism (thyroid
5.23 kPa (NR 4.5–6 kPa), base excess 8.2 mmol/L stimulating hormone 12.51 mIU/L, range 0.32–5.00)
(NR 2 to 2 mmol/L), HCO3 18.0 mmol/L (NR 22– and iron deficiency (iron 2.4 umol/L (NR 12.6–26),
26 mmol/L), lactate 10.3 mmol/L (NR 0.3–0.7)) and Transferrin Saturation 3% (NR 20–45%).
reducing level of consciousness. She required intub- Further examination of the clinical presentation
ation and mechanical ventilation due to progressive and social history revealed a history of mouse infest-
hypoxia and cardiogenic shock, and was commenced ation at her home; no travel history or substance
on noradrenaline. A transthoracic echocardiogram misuse was found. Atypical serology including
revealed dilated left and right ventricles with severely Chlamydia psittaci, Coxiella and Leptospirosis were
reduced biventricular function with an estimated left sent. Hepatitis screen, HIV serology, respiratory
ventricular (LV) ejection fraction of 10% (Simpson’s viral polymerase chain reaction, Pneumococcal and
biplane). She was referred to our cardiac intensive Legionella urinary antigen were all negative, as were
care unit for consideration of veno-arterial extra-cor- the autoimmune (ESR, rheumatoid factor,
poreal membrane oxygenation support (VA-ECMO). Cardiolipin IgG and IgM antibodies, anti-cyclic
She was transferred to our institution convention- citrullinated peptide antibody, Complement C3 and
ally and commenced on ionotropic support with mil- C4, U1RNP, SS-A/Rho, SS-B/La, centromere B,
rinone (0.7 mcg/kg/min). Initial chest radiograph Scl-70, Jo-1, Sm proteins, anti-nuclear antibody), vas-
revealed left basal atelectasis but no focal consolida- culitis (anti-neutrophil cytoplasmic antibody,
tion; electrocardiogram showed sinus rhythm and immunoglobulins G, A, M and E, anti-glomerular
Troponin T was 46 ng/L (normal range ¼ 0–40). basement membrane antibody), myositis (antibodies
Levosimendan was given on day 9 for further to Mi-2, PM/Scl, Ku, PL7, PL12, Jo-1) and myocar-
inotropy, titrated up to 0.2 mcg/kg/min and her nor- ditis screens (Mycoplasma pneumoniae, Enterovirus,
adrenaline dose was subsequently weaned. Our initial Adenovirus, Parechovirus, Human Herpes virus Type
transoesophageal echocardiography (TOE) showed 6 and Influenza A and B).
persistence of severe LV systolic impairment (global Throughout her initial admission, she remained
hypokinesis, worse at inferior and septal walls) with persistently febrile, therefore antimicrobial therapy
an estimated ejection fraction of 30–35%) and severe was escalated to Meropenem and subsequently chan-
right ventricular (RV) systolic impairment with ged to Ceftazidime and Metronidazole. Continuous
elevated RV systolic pressures (estimated right ventri- veno-venous haemodiafiltration with an effluent rate
cle systolic pressure 42 mmHg) and severe tricuspid of 45 mL/kg/h was commenced for intravascular cool-
regurgitation (TR). Supportive care included diuresis ing and renal replacement therapy.
with loop diuretic and empirical broad-spectrum anti- Lumbar puncture was performed which was nega-
biotics (Ciprofloxacin, Gentamicin and Teicoplanin) tive for infection (clear cerebrospinal fluid, glucose
with cover for atypical organisms and fungal infection 5 mmol/L, protein 0.33 g/L, white cell count < 1/
(Caspofungin and Doxycycline). She was not deemed cu.mm, red blood cell 420/cu.mm, no organisms on
a candidate for cardiac transplant due to her high BMI Gram stain and no growth after 48-h incubation). A
of 53.3 kg/m2 and premorbid neurocognitive status percutaneous RV biopsy was also performed 11 days
(low mood with suicidal ideation and agoraphobic into her inpatient stay, which showed morphologic-
behaviour) with lack of social and family support. ally normal myocardium and overlying endocardium,
Concurrently she was not considered a candidate for with no significant inflammation, granuloma forma-
a mechanical assist device, since in the UK it is not tion or malignancy but mainly mature adipose tissue.
considered destination therapy. CT-pulmonary angiog- Congo red stain for amyloidosis and Perls stain for
raphy (CTPA) did not show pulmonary emboli and the haemosiderin was also negative.
coronary arteries were unobstructed on invasive coron- Due to continued raised left atrial pressures, diuresis
ary angiography. was continued with optimisation of her medications for
Initially, she was extubated but due to agitation and biventricular failure. A surgical tracheostomy was per-
neurological impairment the patient was re-intubated. formed for ventilatory weaning and control of her
She developed focal signs of left-sided facial droop, agitation.
dysarthria and reduced left upper limb power. A CT On day 21 of her admission, she was found to be
scan of the brain revealed a right middle cerebral artery Leptospirosis IgM positive. Ceftriaxone was com-
(MCA) territory infarct the aetiology of which was felt menced and Public Health England was notified.
to be cardiac embolus. This was confirmed on subse- Further transthoracic echocardiogram showed some
quent TOE revealed left atrial appendage clot. The improvement in function, but she was still dependent
patient was continued on cardiorespiratory support on inotropic support. She was transferred back to her
and a repeat levosimendan dose was given at seven local hospital 30 days after her admission.
Forbat et al. 353

Biventricular and renal function slowly improved, and survived? Or is the biventricular failure simply a
she was discharged to a medical ward for ongoing by-product of severe sepsis and not leptospirosis spe-
neuro-rehabilitation. She currently mobilises with a cific? The pathophysiology behind cardiac involve-
frame and is able to perform activities of daily living ment beyond myocarditis as the cause of cardiac
independently failure in leptospirosis, has yet to be elucidated.

Authors’ contribution
Discussion
EF and MR wrote the manuscript. All the authors edited
This case has highlighted that a good clinical history the manuscript for intellectual content.
is pertinent to the diagnosis of leptospirosis, on
detailed questioning the patient was in fact living Authors’ note
within squalid conditions of a rodent infested resi- Ethics approval for this case report was not required.
dence. If a detailed social history had not been Written informed consent for the publication of anon-
sought, the diagnosis may have been missed. ymised clinical information was gained from the patient.
Especially given that her presentation was so unusual,
with no literature on biventricular failure in leptospir- Declaration of conflicting interests
osis. Furthermore, a predisposition for leptospirosis The author(s) declared no potential conflicts of interest with
has also been reported in those who suffer with respect to the research, authorship, and/or publication of
chronic alcoholism and smoking1 – both of which this article.
were pertinent in this patient.
There is an abundance of literature on the evidence Funding
of neurological, renal and respiratory failure subse- The author(s) received no financial support for the research,
quent to leptospirosis. Some have reported that car- authorship, and/or publication of this article.
diac involvement is often disguised by pulmonary
haemorrhage or hepatorenal failure,5 however this References
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could this be the reason that the patient in our case