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Case Report
Revised

Case Report: Myocarditis along with fulminant hepatic failure secondary to acute hepatitis E infection

[version 2; peer review: 1 approved with reservations, 1 not approved]
PUBLISHED 18 Nov 2019
Author details Author details
OPEN PEER REVIEW
REVIEWER STATUS

Abstract

Myocarditis, defined as the inflammation of myocardial tissue has many causes which may be viral, metabolic or bacterial in origin. In this case, we report a patient aged 22 years who was admitted with presenting complains of loss of consciousness, generalized muscle rigidity and yellowish discoloration of the skin. During the course of his hospital stay, the patient developed signs of myocarditis and later died of hypotensive shock. Viral serology was positive for the presence of hepatitis E virus (HEV), a rare cause of myocarditis. HEV infection can range from asymptomatic disease course to fulminant hepatitis but in rare cases, it has been found to be a cause of myocarditis. This is so far the sixth case of hepatitis E induced myocarditis.

Keywords

Myocarditis, Hepatitis, hepatic failure, Hepatitis E, Acute hepatitis

Revised Amendments from Version 1

This version 2 contains major changes after considering and carefully working on all the comment from the two assigned reviewers. There were some technical corrections required like those of abbreviations and measurement units. In the comments, some objections were made on the investigations, which are justified, but only an explanation can be given as mentioned in the response to reviewers, since due to the unfortunate demise of the patient, all investigations were not done. Figure 1 was also updated.

See the authors' detailed response to the review by Jun Inoue
See the authors' detailed response to the review by Madhumita Premkumar

Introduction

Hepatitis E virus (HEV) is an important cause of morbidity and mortality and constitutes a significant public health problem1. Hepatitis E transmission includes fecal-oral, vertical and transfusion routes2. Symptoms of HEV infection ranges from asymptomatic to fulminant hepatitis, which is most common in pregnant women. Other extra-hepatic manifestations seen in HEV infection include Guillian Barre syndrome, neuralgic amyotrophy, glomerulonephritis, cryoglobulinemia, pancreatitis, leukemia, thrombocytopenia, meningitis, thyroiditis, neuro-myopathy, vestibular neuritis and arrhythmias3,4. This extra-hepatic involvement seen in an association of HEV is documented in various articles but needs further clinical studies and research3. Here we report a rare case of HEV virus-associated myocarditis in Karachi, Pakistan. The other four reported cases are documented in India5,6. and one case was documented in a western hemisphere traveler7.

Case presentation

A 22-year-old male student, from the Sindh province of Pakistan, unmarried, presented to the emergency department of Jinnah Postgraduate Medical Centre, Pakistan, in April 2018 with loss of consciousness, convulsions, and generalized rigidity. He was admitted to the gastroenterology high dependency unit (HDU). On physical examination his temperature was 100°F, pulse was 136 bpm, blood pressure (BP) of 110/70 mm/Hg, and respiratory rate of 28 breaths per minute. The patient was unresponsive to pain with bilateral reactive pupil, and there was a slightly yellow skin pigmentation of palms and soles with yellow sclera. The patient was not maintaining oxygen saturation hence intubation and ventilatory support were required and so the patient was shifted to the intensive care unit. The abdominal exam was normal without hepatosplenomegaly or ascites, respiratory and heart sounds were audible and normal. His past medical history was negative and was non-contributory. Laboratory parameters at that time revealed, altered liver function tests (LFTs) with Alanine Aminotransferase (ALT) 1160 u/L (reference range, 7–56 u/L); aspartate aminotransferase (AST) 225 u/L (reference range, 10–40 u/L); gamma-glutamyltransferase (GGT) 51 u/L (reference range, 9–50 u/L); alkaline phosphatase (ALP) 372 u/L (reference range, 150–480 u/L); serum albumin 4.2 g/dL (reference range, 3.5–5.5 g/dL); total bilirubin 4 mg/dL (reference range, 0.1–1.2 mg/dL); Prothrombin time (PT) 13 seconds (reference range, 11–14 seconds); and international normalized ratio (INR) 1.13 (reference range, 0.9–1.2). His blood urea was 61 mg/dL (reference range, 7–20 mg/dL), serum creatinine of 1.64 (reference range, 0.6–1.2 mg/dL), with a normal serum electrolyte panel. A viral screen of the patient's blood was ordered and HEV IgM and IgG was positive, with negative serology for Hepatitis A, Hepatitis B, Hepatitis C, Hepatitis D, Dengue, Typhoid, Cox-B, Epstein-Barr virus (EBV), Leptospira, Herpes simplex virus, Adenovirus, and HIV. Blood cultures were negative, rapid malaria test was negative, labs for antinuclear antibody, anti-mitochondrial antibody, and anti-smooth muscle antibody was also negative. Sonographic investigation showed normal abdominal structures. Patient’s blood urea and creatinine returned to normal on the second day of admission and LFTs started recovering by the fourth day. On the seventh day of hospitalization, the patient had normal total bilirubin with slightly raised LFTs of ALP 120 u/l, ALT 115 u/L, AST 62 u/L, GGT 68 u/L and platelet of 201×109/L, leukocytes 27×109/L with high-grade fever. On the seventh day of admission, the patient became hypotensive with BP of 92/68 mm/Hg and a pulse of 148. Physical examination revealed distended external jugular veins, with bilateral pedal edema and bilateral chest congestion. Chest examination showed decreased respiratory sounds from the right and left lung base with muffled heart sounds. On further investigations and workups, a chest X-Ray showed bilateral pulmonary congestion at the base of the lung with cephalization of vessels. Electrocardiography (ECG) showed nonspecific ST-segment and T-wave abnormalities with supraventricular tachycardia (Figure 1). Creatine phosphokinase (CPK) came out to be 8414 u/l (the patient had no history of seizures). Coronary angiography was performed to exclude acute coronary syndrome, which revealed no obstructive lesion in the coronary artery. Echocardiography was performed which demonstrated impaired left ventricular function with diffuse hypokinesia without any pericardial effusion and an ejection fraction of 30 %. The sign and symptoms together with workups and labs were highly suggestive of myocarditis. The patient was treated accordingly an injection of Omeprazole (40 mg once daily), 25% dextrose (as per needed), Colistimethate sodium (2 million IU thrice daily) and moxifloxacin (400 mg once daily), acyclovir (500 mg thrice daily); syrup lactulose (30 ml 6 hourly) was given nasogastrically; and patient was kept sedated with propofol. Despite aggressive medical support, the patient abruptly developed cardiopulmonary arrest on the eighth day of admission, resuscitation was performed but the patient didn’t recover and died of cardiopulmonary arrest. More specific investigations were planned but could not be performed post-mortem.

8993bb56-80b2-4066-b7b1-beeda68b659b_figure1.gif

Figure 1. Electrocardiogram of the patient.

Discussion

According to the World Health Organization, the annual incidence of HEV infection is 20 million around the globe, with the majority of cases from East and South Asia8. It is common in resource-limited countries with limited access to essential water, sanitation and health services. Our patient contracted an acute HEV infection causing acute fulminant hepatitis leading to hepatic encephalopathy, coma, and loss of consciousness. He was intubated and managed appropriately in intensive medical care, patients LFT’s were recovering but unfortunately, he developed myocarditis and died of cardiopulmonary arrest. Myocarditis is the inflammation of the myocardium with inflammatory infiltrate, necrosis, and degeneration of myocytes in the myocardium8. Among all other causes of myocarditis, viral etiology appears to be the most common in developed countries, whereas, in developing countries, rheumatic carditis, Chagas disease, and HIV associated diseases are more common9,10. As there is so much clinical variability, diagnosing myocarditis is a challenge. Diagnostic guidelines such as those of the American college of cardiology/American heart association and Dallas criteria provide help with early diagnosis4. On eighth hospitalization day, our patient developed hypotension and distended jugular venous distension, pedal edema, bilateral pulmonary edema, and muffled heart sounds. On initial workup, cardiac enzymes, ECG, CPK tests were planned. Echocardiography is recommended as an initial diagnostic tool for suspected myocarditis11. According to small observational studies, it was suggested that magnetic resonance imaging (MRI) can identify morphological and anatomical changes such as inflammation, edema and myocyte injury in the myocardium. Due to myocardial inflammation, T1 and T2 relaxation times and spin densities give accurate tissue characterization12. Endomyocardial biopsy is also an important diagnostic tool in myocarditis and sometimes is the only means of diagnosing myocarditis. Endomyocardial biopsy may be used for patients whose condition does not respond to conventional supportive therapy, a patient with acute dilated cardiomyopathy associated with hemodynamic compromise, refractory cases, patients with life-threatening arrhythmias and high-grade AV block13. Histological evaluation is also diagnostic for myocarditis, however, because of variable observer interpretations and focal inflammation sampling error can be high14. In our case, the diagnosis was limited as these confirmatory tests just mentioned i.e. echocardiography, biopsy, and MRI, could not be performed due to the early death of the patient.

Consent

Written informed consent for publication of their clinical details was obtained from the relatives of the patient.

Data availability

All data underlying the results are available as part of the article and no additional source data are required.

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Version 2
VERSION 2 PUBLISHED 05 Feb 2019
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CITE
how to cite this article
Rai L, Salam O, Yaqoob U and Zubair U. Case Report: Myocarditis along with fulminant hepatic failure secondary to acute hepatitis E infection [version 2; peer review: 1 approved with reservations, 1 not approved]. F1000Research 2019, 8:155 (https://doi.org/10.12688/f1000research.17797.2)
NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article.
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Open Peer Review

Current Reviewer Status: ?
Key to Reviewer Statuses VIEW
ApprovedThe paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approvedFundamental flaws in the paper seriously undermine the findings and conclusions
Version 2
VERSION 2
PUBLISHED 18 Nov 2019
Revised
Views
8
Cite
Reviewer Report 02 Dec 2019
Jun Inoue, Division of Gastroenterology, Tohoku University Graduate School of Medicine, Sendai, Japan 
Approved with Reservations
VIEWS 8
The authors modified most parts that were commented by reviewers, but did not respond to comment no. 7. There are several reported cases of HEV infection who developed myocarditis. Please discuss the clinical characters of such patients including this report. Are ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Inoue J. Reviewer Report For: Case Report: Myocarditis along with fulminant hepatic failure secondary to acute hepatitis E infection [version 2; peer review: 1 approved with reservations, 1 not approved]. F1000Research 2019, 8:155 (https://doi.org/10.5256/f1000research.23395.r56683)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
Views
9
Cite
Reviewer Report 02 Dec 2019
Madhumita Premkumar, Institute of Liver and Biliary Sciences, Chandigarh, India 
Not Approved
VIEWS 9
Although the authors have revised their manuscript, I'm afraid there are too many confounding elements to definitely say this this viral myocarditis. In most cases it is superadded sepsis that leads to septic cardiomyopathy. It can be labeled possible myocarditis ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Premkumar M. Reviewer Report For: Case Report: Myocarditis along with fulminant hepatic failure secondary to acute hepatitis E infection [version 2; peer review: 1 approved with reservations, 1 not approved]. F1000Research 2019, 8:155 (https://doi.org/10.5256/f1000research.23395.r56682)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
Version 1
VERSION 1
PUBLISHED 05 Feb 2019
Views
21
Cite
Reviewer Report 08 Aug 2019
Madhumita Premkumar, Institute of Liver and Biliary Sciences, Chandigarh, India 
Not Approved
VIEWS 21
The case report describes a 22- year old patient who had acute viral Hepatitis E who presented with altered sensorium possibly hepatic encephalopathy, but there are several unexplained details which alter the diagnostic specificity:
  1. Why did
... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Premkumar M. Reviewer Report For: Case Report: Myocarditis along with fulminant hepatic failure secondary to acute hepatitis E infection [version 2; peer review: 1 approved with reservations, 1 not approved]. F1000Research 2019, 8:155 (https://doi.org/10.5256/f1000research.19457.r51461)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
  • Author Response 18 Nov 2019
    Uzair Yaqoob, Dow University of Health Sciences, Karachi, Pakistan
    18 Nov 2019
    Author Response
    (Responses numbered acc to comments)
    1.Patient was in hepatic encephalopathy and was showing signs of raised ICP due to which muscle tone was 
    increased and muscle rigidity was present.
    2. ... Continue reading
COMMENTS ON THIS REPORT
  • Author Response 18 Nov 2019
    Uzair Yaqoob, Dow University of Health Sciences, Karachi, Pakistan
    18 Nov 2019
    Author Response
    (Responses numbered acc to comments)
    1.Patient was in hepatic encephalopathy and was showing signs of raised ICP due to which muscle tone was 
    increased and muscle rigidity was present.
    2. ... Continue reading
Views
23
Cite
Reviewer Report 15 Jul 2019
Jun Inoue, Division of Gastroenterology, Tohoku University Graduate School of Medicine, Sendai, Japan 
Not Approved
VIEWS 23
In this paper, Rai et al. described a patient with acute hepatitis E virus infection who developed acute heart failure. Some cases with hepatitis E virus-associated myocarditis have been reported previously and the authors considered the development of myocarditis also in ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Inoue J. Reviewer Report For: Case Report: Myocarditis along with fulminant hepatic failure secondary to acute hepatitis E infection [version 2; peer review: 1 approved with reservations, 1 not approved]. F1000Research 2019, 8:155 (https://doi.org/10.5256/f1000research.19457.r50664)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
  • Author Response 05 Nov 2019
    Uzair Yaqoob, Dow University of Health Sciences, Karachi, Pakistan
    05 Nov 2019
    Author Response
    Thank you so much for taking your time out and reviewing our work. we have worked and updated our article according to your comments and it will soon be online. ... Continue reading
  • Author Response 18 Nov 2019
    Uzair Yaqoob, Dow University of Health Sciences, Karachi, Pakistan
    18 Nov 2019
    Author Response
    (Response to rest of the comments)
    1.as patient was on mechanical ventilation, he had dyspnoea, became tachycardic with raised JVP, ECG got done showed ST-T wave abnormalities along with SVT, ... Continue reading
COMMENTS ON THIS REPORT
  • Author Response 05 Nov 2019
    Uzair Yaqoob, Dow University of Health Sciences, Karachi, Pakistan
    05 Nov 2019
    Author Response
    Thank you so much for taking your time out and reviewing our work. we have worked and updated our article according to your comments and it will soon be online. ... Continue reading
  • Author Response 18 Nov 2019
    Uzair Yaqoob, Dow University of Health Sciences, Karachi, Pakistan
    18 Nov 2019
    Author Response
    (Response to rest of the comments)
    1.as patient was on mechanical ventilation, he had dyspnoea, became tachycardic with raised JVP, ECG got done showed ST-T wave abnormalities along with SVT, ... Continue reading

Comments on this article Comments (0)

Version 2
VERSION 2 PUBLISHED 05 Feb 2019
Comment
Alongside their report, reviewers assign a status to the article:
Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions
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