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Case Report
Revised

Case Report: Complete heart block as a manifestation of cardiac metastasis of oral cancer

[version 2; peer review: 2 approved]
PUBLISHED 11 Nov 2020
Author details Author details
OPEN PEER REVIEW
REVIEWER STATUS

Abstract

Metastatic tumors of the heart presenting with complete heart block (CHB) is an extremely uncommon case. There are no available guidelines in managing CHB in terminal cancer. Permanent pacemaker implantation in such cases is a challenge in terms of clinical utility and palliative care.
We report a case of a 24-year-old man suffering from tongue cancer presenting with CHB. An intracardiac mass and moderate pericardial effusion were present, presumed as the metastatic tumor of tongue cancer. We implanted a temporary pacemaker for his symptomatic heart block and cardiogenic shock, and pericardiocentesis for his massive pericardial effusion. We decided that a permanent pacemaker would not be implanted based on the low survival rate and significant comorbidities.
Multiple studies report a variable number of cardiac metastasis incidence ranging from 2.3% to 18.3%. It is rare for such malignancies to present with CHB. The decision to implant a permanent pacemaker is highly specific based on the risks and benefits of each patient. It needs to be tailored to the patient’s functional status, comorbid diseases, prognosis, and response to conservative management.

Keywords

tongue cancer, cardiac metastasis, complete heart block, case report

Revised Amendments from Version 1

We added a clearer definition of improvement in general condition; supportive treatment including dopamine use; and the reason of TPM extraction. We also added our suggestion of TPM use in such cases.

See the authors' detailed response to the review by Radityo Prakoso

Introduction

Cardiac metastasis is the least common presentation in malignant cancer. Primary cardiac tumors are also rare (on postmortem analysis, commonly between 0.01% to 0.1%). However, the frequency of secondary metastatic tumors to the pericardium, myocardium, great vessels, and coronary arteries are between 0.7% to 3.5% in the general population and up to 9.1% in patients with a history of malignancies1.

Complete heart block (CHB) as the primary clinical presentation of heart metastasis is very unusual2. There are currently no guidelines for the management of CHB in terminal stage of cancer.

We report a case of CHB caused by cardiac metastasis and review the literature to further help the management of our patient.

Case presentation

A 24-year old Asian man was admitted to the cardiology department with CHB and hypotension. The patient was a chef and had a history of tongue cancer for six months, and had undergone 30 cycles of radiotherapy. A week before, the patient came to the emergency department (ED) because of oral bleeding and general weakness. The patient denied any history of cardiovascular disease.

The patient was presented with chest discomfort and general weakness. He was hypotensive and bradycardic with a blood pressure of 80/40 mmHg, regular heart rate of 44 beats per minute, respiratory rate of 18 breaths per minute, and oxygen saturation of 97% on room air. Chest auscultation was clear, and no murmurs heard. Electrocardiogram showed CHB with a junctional escape rhythm at 44 bpm (Figure 1). Echocardiography showed normal left ventricle kinetic, normal left ventricular ejection fraction (62%), and normal right ventricle systolic function. There were moderate pericardial effusion and intracardiac masses (2.1 × 0.9 cm and 1.8 × 0.8 cm) in the right atrial and septal leaflet of tricuspid. Hyperechoic areas in the annulus of tricuspid, lateral wall of right atrium and right ventricle, and interventricular septum were also found in an echocardiogram (Figure 2 and Figure 3). Laboratory finding revealed anemia (hemoglobin 8.5 g/dL; normal range 13.3-16.6 g/dL), leukocytosis (white blood count 18,470/mL; normal range 3,370-10,000/mL), hypoalbuminemia (albumin 2.6 g/dL; normal range 3.4-5.0 g/dL), hypokalemia (potassium 3.4 mmol/L; normal range 3.5-5.1 mmol/L), and hypercalcemia (calcium 16.2 mg/dL, corrected calcium 16.9 mg/dL; normal range 8.6-10.3 mg/dL).

68c843aa-1eeb-46f7-b013-9c338ee6cd7b_figure1.gif

Figure 1. Electrocardiography on the first day of consultation showed complete heart block.

68c843aa-1eeb-46f7-b013-9c338ee6cd7b_figure2.gif

Figure 2. Echocardiography showed the presence of a mass in the right atrium and septal leaflet of tricuspid (yellow arrow).

Hyperechoic areas were found in the annulus of tricuspid, lateral wall of right atrium and right ventricle, and interventricular septum (red arrows).

68c843aa-1eeb-46f7-b013-9c338ee6cd7b_figure3.gif

Figure 3.

Pericardial effusion was found in (A) anterior, posterior, (B) inferior, (C) base, and (D) left-lateral of the heart.

Previous magnetic resonance imaging (MRI) of head and neck, six months before this admission, revealed malignant tongue mass (staging AJCC 2010 of lip and oral cavity mass: T4N1Mx) and bilateral nasal cavity thickening. Multi-slice computed tomography (CT) scan of the head revealed an enhancing mass 1×1.3×1.5 cm at the base of the tongue and multiple lymph node enlargements subcentimeter in the upper and lower paratracheal. Histopathology examination of tongue biopsy confirmed poorly differentiated squamous cell carcinoma.

A temporary pacemaker was immediately implanted as the patient showed symptomatic heart block and cardiogenic shock. After general supportive treatment, including intravenous dopamine administration, electrolyte imbalance correction, supportive treatment of general weakness condition, anemia, hypoalbuminemia, and infection, the patient showed improvement in general condition. No hemodynamic instability was observed when temporary pacemaker was turned off. However, the CHB persisted despite electrolyte imbalance correction.

Multidiscipline team discussions involving electrophysiologist, otolaryngologist, and internist, resulted in decision to focus more on palliative care for this patient. Considering the poor prognosis of this cancer, risk of permanent pacemaker (PPM) implantation, and severe comorbidities, we decided not to implant a PPM after acquiring the patient’s and his family’s consent.

Considering the risk of infection in prolong use of temporary pacemaker, on the 7th day the temporary pacemaker was extracted.

On the 14th day of admission, the patient developed pleural effusion, and worsening pericardial effusion with echocardiogram showed massive pericardial effusion and sign of tamponade. Further chest X-ray evaluation on the 14th day showed left parahilar ground glass appearance with suspicions of lung metastasis and pleural effusion (Figure 4). Pericardiocentesis was then performed with pericardial fluid showing hemorrhagic typical for malignant disease.

68c843aa-1eeb-46f7-b013-9c338ee6cd7b_figure4.gif

Figure 4. Chest X-ray on the 14th day of treatment showed left parahilar ground glass appearance with suspicion of lung metastasis and pleural effusion.

Pericardial fluid pigtail was already inserted for drainage of pericardial effusion.

The patient died due to respiratory failure and septic shock 20 days after showing the first symptoms of cardiac metastasis.

Discussion

From the literature, we acquired a total of 14 articles regarding heart block as a manifestation of cardiac metastasis, as summarized in Table 1215. Oral cavity, uterus, and thyroid are the most common primary cancers that metastasize to the heart. Squamous cell carcinoma was the primary histologic finding. Heart metastasize may be present with clinically silent symptoms to an alarming presentation of hemoptysis and syncope. Locations of metastasis were mostly in the right ventricle, supporting the hypothesis of the hematologic spread of cancer cells. PPM implantation was performed in 10 cases, yet only one case reported a significant lifespan after PPM implantation.

Table 1. Literature review of case reports regarding heart block caused by cardiac metastasis.

ArticleAge
(years)
Primary site
of cancer
Type of cancer
cell
Location
of cardiac
metastasis
Sign /
symptoms
ECG findingsOther cardiac
manifestation
PPM
implantation
Type of PPMReversibility
of heart
block
Treatment of
cardiac metastasis
Diagnostic
tools to
detect cardiac
metastasis
Lifespan
after cardiac
manifestation
Buckberg
and Fowler,
19613
42bronchogenic
carcinoma
adeno carcinoma interventricular
septum,
anterior wall of
left ventricle
shortness of
breath, cough
with blood
complete heart
block
intracardiac
mass
no No no none postmortem
autopsy
6 weeks
Clifford
et al., 20038
64lymphfollicular small
cell cleaved
lymphoma
interventricular
septum and RV
anterior wall
nausea,
diaphoresis,
and dizziness
complete heart
block with a
ventricular
escape
RV massyesdual chamberyeschemotherapyechocardiogram2 years, still
alive
Mocini
et al., 20059
44lungmalignant
neoplasm
interventricular
septum, left
ventricular wall
syncopecomplete heart
block
mild pericardial
effusion
yesdual chamberno none postmortem
autopsy
3 days
Ferraz
et al.,
200610
63uterussquamous cell
carcinoma
interventricular
septum and RV
fatigue and
dyspnea on
mild exertion
complete heart
block
pericardial
effusion, RV
mass
yespermanent
atrioventricular
epimyocardial
pacemaker
nosurgery for RV massechocardiogram,
CT
4 months
Ozyuncu
et al.,
200611
56right thighmalignant
melanoma
interventricular,
interatrial
septum
nausea,
vomiting,
presyncope
complete heart
block
mitral
regurgitation,
pericardial
effusion
yes VDDnochemo-
immunotherapy
CT thorax,
echocardiogram
2 months, still
alive
Knowles
et al.,
200712
42right
maxillary
sinus
right
maxillary
sinus
not statedsyncopecomplete heart
block with a
ventricular
escape
pericardial
effusion
nonoyeschemotherapyechocardiogram,
CT
1 year
Rathi et al.,
200813
67skinmalignant
melanoma
all myocardial
walls
asymptomaticcomplete heart
block,
junctional
escape
pericardial
effusion
nonointermittent
complete
heart block
chemotherapyechocardiogram,
MRI
not stated
Lin et al.,
201514
74thyroidpapillary thyroid
carcinoma
RVOTexertional
dyspnea and
palpitations
first-degree
atrioventricular
block
and
subsequently
a new
intermittent
complete
atrioventricular
block
RVOT massyesnot statednononeCTA, C MRI,
PET Scan
not stated
(not long after
discharge)
Yoneda
et al.,
201615
53gingivalsquamous cell
carcinoma
atrial septum,
left ventricle,
AV node
cough, syncopecomplete
heart block,
ventricular
fibrillation
noneno No nochemotherapypostmortem
autopsy
4 weeks
Park et al.,
20164
54right legleiomyosarcomainterventricular
septum
dizziness and
dyspnea
complete heart
block and
idioventricular
escaped
rhythm of
bifascicular
block
morphology
VT,
intraventricular
mass
yesdual chambernopalliative
chemotherapy
MRI3 months
Yoshihiro
et al., 20175
57thyroidsquamous cell
carcinoma
interventricular
septum
cough and
shortness of
breath
complete heart
block
RV massyesnot statednochemoradiotherapyFDG-PET25 days
Kansai
et al., 20076
56lungadenocarcinomainterventricular
septum
dull pain,
presyncope
complete heart
block
LV massyesnot statednononeechocardiogram,
CT, postmortem
autopsy
19 days
Kumar
et al., 20182
28tonguesquamous cell
carcinoma
interventricular
septum
syncopecomplete heart
block
noneyesnot statednononePET Scan5 days
Cho et al.,
20187
70oral cavitysquamous cell
carcinoma
interventricular
septum
dizzinesscomplete heart
block with a
ventricular
escape
none yes DDDnopalliative
chemotherapy
FDG PETnot stated

The prevalence of cardiac metastasis, in general, is arguably low. However, multiple studies reported a variable number of cardiac metastasis incidence ranging from 2.3% to 18.3%. The prevalence of malignancy originating from the oral cavity was 5.3%. The involvement of pericardium made up two-thirds of all cardiac metastasis. Myocardium and endocardium involvement each made up one-third of all cardiac metastasis. Only 5% involved the endocardium. The most common site of metastasis for squamous cell carcinomas is epicardium (41.4%)16.

Myocardial infiltration by cancer cells may present with arrhythmias, such as atrial flutter or fibrillation, premature beats, or ventricular arrhythmias. Conduction system involvement may induce a various degree of atrioventricular blocks16.

The presence of right atrial mass in our patient supports the possibility of hematologic spreading of metastatic cancer cells into the endocardium. Pericardial effusion may represent metastasize or inflammatory reaction toward the malignancy. The presence of CHB suggests the infiltration of the heart conduction system.

Valves are an uncommon site for metastasis because of the absence of vessels in the physiological valvular stroma and the constant cusp motion. Bussani et al. reported, out of over a thousand of post-mortem examination, there was only one case of valve involvement16. The mass in the septal leaflet of tricuspid valves appeared in echocardiography examination in this patient showed valve involvement of cardiac metastasis.

Asymptomatic in the early stages, cardiac metastasis could lead to a wide range of signs and symptoms, such as cardiac failure, conduction disturbances, angina, and pain as it progresses. Disruption of the cardiac conduction system by cardiac metastases can lead to lethal arrhythmias, including atrial fibrillation with a rapid ventricular response, CHB, or ventricular fibrillation1. From the literature, we acquired 14 cases reporting CHB as a manifestation of cardiac metastasis originating from various malignancies, three of which are from the oral cavity.

Cardiac masses in our case presented with features favoring tumor, such as echo density similar to myocardium, normal wall motion, valvular lesion, no history suggestive of coronary artery disease, and a clinical history of oral cancer as primary site suspected to metastasize to the heart.

Cardiac MRI is the best imaging modality and, along with positron emission tomography scanning, are mostly used in investigating the extent of infiltration by malignant cells2,17. Patients with specific cardiac devices, such as pacemaker and defibrillators, would be disqualified from undergoing MRI, an important consideration given the frequency at which arrhythmia complicates cardiac metastasis17. The temporary pacemaker implanted during the early stages of CHB made MRI impractical for our patient.

In our patient, CHB was initially thought to be the result of electrolyte imbalance. However, as the electrolyte was restored to its normal level without any improvement of CHB, it suggested that CHB was caused by infiltration of the metastatic cell to the conduction system of the heart. The presence of hemorrhagic pericardial effusion supports the suggestion of pericardial metastasis. Regardless of the lack of histological confirmation, we suggest that this case was cardiac metastasis diagnosed antemortem.

Predominantly, after a reversible or transient cause of bradycardia is excluded, cardiac pacing indication is decided by bradycardia severity instead of its etiology. The European Society of Cardiology (ESC) guidelines state that some types of persistent bradycardia require permanent pacing. In acquired AV block, pacing is indicated in patients with second-degree type 2 or third-degree AV block regardless of symptoms (class I)18. Similar to the ESC guidelines, the American College of Cardiology/American Heart Association/Heart Rhythm Society guidelines also state that patients with transient or reversible causes of AV block should receive medical and supportive treatment if necessary, including temporary transvenous pacing, prior to confirmation of the need for permanent pacing (class of Recommendation/COR I). In addition, for patients with acquired CHB not associated with physiologic or reversible etiology, permanent pacing is recommended regardless of symptoms (COR I)19.

In our case, we assume that the CHB was persistent after trying to correct possible external causes, such as hypercalcemia and hypokalemia, and after a sufficient waiting period. Moreover, it is still also unknown whether CHB would resolve after cancer treatment2,20. Our literature review showed that there are only two cases of metastatic CHB that are reversible after undergoing chemotherapy for cardiac metastasis8,12, while several other cases showed that CHB is irreversible27,911,1315. The short life expectancy in patients with metastatic CHB also makes it difficult to follow up on CHB reversibility. Our literature review showed only one metastatic CHB case reported a significant lifespan more than two years after PPM implantation8, while other cases reported a lifespan of no longer than one year27,915.

Our patient was expected to continue the radiotherapy cycle. Radiotherapy (RT) itself can induce pacemaker malfunction. Software impairments are the primary manifestation of malfunction during RT. This will perhaps lead to pacemaker reset, leaving only device basic function. Radiation dosage appears to contribute less to inducing pacemaker malfunctions than the beam energy of the RT21.

For patients with indications for permanent pacing but accompanied with significant comorbidities or who are anticipated having a shortened lifespan because of terminal progressive illness, the implantation of a PPM should not be performed if it is unlikely to deliver significant clinical benefits or if it hinders the main therapy for the patient’s goal of care. Even though pacemaker implantation risks are rather low, the risk-benefit ratio is not favorable if the possible benefit is also quite low19. Thus, after discussing with our patient and his family, we decided not to implant a PPM after acquiring the patient’s and his family’s consent.

Conclusion

CHB in a patient with oral cancer should increase the physician's suspicion of cardiac metastasis. It is rare for such malignancies to present with CHB. Temporary pacemaker should be considered for patients presenting with CHB and unstable hemodynamic before deciding permanent pacemaker implantation. The decision to implant a permanent pacemaker is highly specific based on the risks and benefits of each patient. It needs to be tailored to the patient’s functional status, comorbid diseases, prognosis, and response to conservative management. Even though the pacemaker implantation risks are rather low, the risk-benefit ratio is not favorable if the possible benefit is also quite low. A permanent pacemaker was not implanted in our patient because of the poor prognosis, severe comorbidities, and low expected lifespan.

Consent

Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient’s parent.

Data availability

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

Comments on this article Comments (1)

Version 2
VERSION 2 PUBLISHED 11 Nov 2020
Revised
Version 1
VERSION 1 PUBLISHED 15 Oct 2020
Discussion is closed on this version, please comment on the latest version above.
  • Reader Comment 03 Feb 2021
    Mohammad Saifur Rohman, Cardiology and vascular medicine, faculty of medicine, Brawijaya University, Malang, Indonesia
    03 Feb 2021
    Reader Comment
    Very interesting and might be considered to be indexed.
    This is an extremely rare case of complete heart block which might related or not to tongue cancer metastatic. Since, no histopathology ... Continue reading
  • Discussion is closed on this version, please comment on the latest version above.
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Andrianto A, Mulia EPB, Suwanto D et al. Case Report: Complete heart block as a manifestation of cardiac metastasis of oral cancer [version 2; peer review: 2 approved]. F1000Research 2020, 9:1243 (https://doi.org/10.12688/f1000research.26438.2)
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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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 11 Nov 2020
Revised
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Reviewer Report 19 Nov 2020
Pieter A.F.M. Doevendans, Department of Cardiology, Experimental Cardiology Laboratory, Division Heart and Lungs, University Medical Center Utrecht, Utrecht, The Netherlands 
Approved
VIEWS 20
General 
Interesting case report, there are only text and grammar issues. Well written, with supporting images. The case deals with a young adult treated for oral carcinoma who developed a symptomatic total AV block. The block persisted after restoration of the electrolyte dysbalance ... Continue reading
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Doevendans PAFM. Reviewer Report For: Case Report: Complete heart block as a manifestation of cardiac metastasis of oral cancer [version 2; peer review: 2 approved]. F1000Research 2020, 9:1243 (https://doi.org/10.5256/f1000research.30564.r74285)
NOTE: 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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Reviewer Report 19 Nov 2020
Radityo Prakoso, Department of Cardiology and Vascular Medicine, Faculty of Medicine, Universitas Indonesia, Jakarta, Indonesia 
Approved
VIEWS 11
I think the author has revised all the suggestions and there ... Continue reading
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Prakoso R. Reviewer Report For: Case Report: Complete heart block as a manifestation of cardiac metastasis of oral cancer [version 2; peer review: 2 approved]. F1000Research 2020, 9:1243 (https://doi.org/10.5256/f1000research.30564.r74597)
NOTE: 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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Reviewer Report 27 Oct 2020
Radityo Prakoso, Department of Cardiology and Vascular Medicine, Faculty of Medicine, Universitas Indonesia, Jakarta, Indonesia 
Approved with Reservations
VIEWS 33
Some typos and grammars should be evaluated. In the presentation, what does author mean about "improvement"? Why did TPM extracted by improvement but the complete heart block was actually not improving? I mean its 7th days were they sure that the ... Continue reading
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CITE
HOW TO CITE THIS REPORT
Prakoso R. Reviewer Report For: Case Report: Complete heart block as a manifestation of cardiac metastasis of oral cancer [version 2; peer review: 2 approved]. F1000Research 2020, 9:1243 (https://doi.org/10.5256/f1000research.29188.r73101)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
  • Author Response 11 Nov 2020
    Andrianto Andrianto, Department of Cardiology and Vascular Medicine, Airlangga University - Dr. Soetomo General Hospital, Surabaya, 60286, Indonesia
    11 Nov 2020
    Author Response
    Some typos and grammars should be evaluated. In the presentation, what does author mean about "improvement"? Why did TPM extracted by improvement but the complete heart block was actually not improving? ... Continue reading
COMMENTS ON THIS REPORT
  • Author Response 11 Nov 2020
    Andrianto Andrianto, Department of Cardiology and Vascular Medicine, Airlangga University - Dr. Soetomo General Hospital, Surabaya, 60286, Indonesia
    11 Nov 2020
    Author Response
    Some typos and grammars should be evaluated. In the presentation, what does author mean about "improvement"? Why did TPM extracted by improvement but the complete heart block was actually not improving? ... Continue reading

Comments on this article Comments (1)

Version 2
VERSION 2 PUBLISHED 11 Nov 2020
Revised
Version 1
VERSION 1 PUBLISHED 15 Oct 2020
Discussion is closed on this version, please comment on the latest version above.
  • Reader Comment 03 Feb 2021
    Mohammad Saifur Rohman, Cardiology and vascular medicine, faculty of medicine, Brawijaya University, Malang, Indonesia
    03 Feb 2021
    Reader Comment
    Very interesting and might be considered to be indexed.
    This is an extremely rare case of complete heart block which might related or not to tongue cancer metastatic. Since, no histopathology ... Continue reading
  • Discussion is closed on this version, please comment on the latest version above.
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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