Pericardial cyst as a cause of recurrent hiccups: a case report

Introduction

Pericardial cysts may be caused by a congenital defect in the pericardial sac, leading to a fluid filled diverticulum which can close off to form a cyst. They constitute approximately 6% of mediastinal masses (1). Often, they are found incidentally, and can be asymptomatic. If symptomatic, this is generally via mass effect on surrounding structures. This can include cardiac compression causing chest pain or congestive heart failure symptoms, pulmonary compression causing cough, dyspnea, or recurrent respiratory infections, or dysphagia due to esophageal compression (2-6). We report a case of a pericardial cyst found incidentally as a cause of persistent hiccups. There are few existing cases of pericardial cysts or other cystic lesions (including hydatid cysts and pleuro-pericardial lymphocele) causing hiccups seen in the literature (7-9). These cases of hiccups are hypothesized to be resulting from mediastinal compression of the phrenic nerve and were treated with open surgical approaches (7-9). We are unable to find any existing cases of pericardial cysts causing persistent hiccups resolved via a video-assisted thoracoscopic surgery (VATS) approach. We present this case in accordance with the CARE reporting checklist (available at https://vats.amegroups.com/article/view/10.21037/vats-23-77/rc).

Case presentation

A 51-year-old Caucasian male presented to outpatient thoracic surgery clinic with a history of persistent, intermittent hiccups for 3 months. He had no other notable symptoms. The patient’s past medical history was otherwise non-contributory. He had no allergies or home medications at the time of onset. He is a non-smoker. The patient had previous minor unrelated surgeries. His family history includes arrhythmogenic right ventricular cardiomyopathy, for which he has been monitored by cardiology with echocardiography, cardiac magnetic resonance imaging (MRI), and myocardial perfusion imaging. On physical exam, there were no pertinent findings.

He was first seen by his family physician, who completed a history and physical exam. There was no clear inciting factor or physical exam findings, however, at the time, the patient had recently undergone dental surgery after which the hiccups seem to have begun. They occurred several times per day, lasting 15 to 20 minutes in duration before subsiding. He returned to his dentist for care and to have sutures removed, which did not decrease his hiccups. His family physician first prescribed chlorpromazine (50 mg four times daily), which was unsuccessful in treating his hiccups. He was then started on a trial of pregabalin (75 mg three times daily), which provided only partial relief. A chest X-ray was performed which showed an irregularity of the right heart border with a normally positioned right hemidiaphragm. Chest computed tomography (CT) revealed a simple fluid filled pericardial cyst of up to 5.3 cm along the lateral aspect of the right atrium (Figure 1). Chest MRI was also completed, which showed a 4.8 cm × 2.7 cm × 4.1 cm benign appearing pericardial cyst within the parietal pericardial recess, abutting the right cardiophrenic space in close proximity to the right phrenic nerve. He was therefore referred to thoracic surgery. After consultation, a trial off pregabalin was completed, resulting in the full return of hiccups. Given the lack of any other potential cause, after a discussion of the risks and benefits of surgery, the patient desired to proceed with resection of the pericardial cyst.

Figure 1 Chest computed tomography scan (coronal view) showing a pericardial cyst (red arrow) adjacent to the right atrium.

Under general anesthesia, a double lumen endotracheal tube was inserted, and position was verified with flexible bronchoscopy. The patient was placed in the left lateral position and four incisions were made over the right lateral chest wall. A multi-port right video-assisted thoracoscopy was performed. An approximately 5 cm by 3 cm pericardial cyst was identified, anterior to the right phrenic nerve (Figure 2). Superiorly, there was a connection to the pericardium. This was dissected off the right phrenic nerve and removed. The remainder of the operation was uncomplicated. Final pathology showed a mesothelium-lined cyst with fibro-adipose wall and chronic inflammation, consistent with a benign pericardial cyst (Figure 3).

Figure 2 Intraoperative thoracoscopic images of pericardial cyst resection via multi-port right VATS. (A) Pericardial cyst (black arrow) situated in the right cardiophrenic space; (B) retraction of the pericardial cyst (black arrow) to reveal the right phrenic nerve (yellow arrow) in close proximity posteriorly. VATS, video-assisted thoracoscopic surgery.

Figure 3 Histopathological images of the resected pericardial cyst. (A) Hematoxylin and eosin stain at 25× magnification, showing a simple cyst with a wall composed of fibroadipose tissue and scattered foci of chronic inflammation; (B) hematoxylin and eosin stain at 200× magnification, showing the lining of the cyst to be composed of mesothelial cells displaying reactive changes, due to chronic lymphoplasmacytic inflammation.

Post-operatively, the patient had an uneventful course. He was discharged home on post-operative day one. Upon discharge, the hiccups had dissipated. Post-operative chest X-ray showed a normally positioned right hemidiaphragm, indicating a functioning right phrenic nerve. He was then seen in clinic for follow-up at 4 and 8 weeks, with complete dissipation of hiccups, and an unremarkable chest X-ray at 4 weeks.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

Pericardial cysts have an incidence of approximately 1 in 100,000 patients, often found incidentally on imaging and typically asymptomatic (5). They are most commonly caused by congenital weakness in the pericardial sac, leading to a diverticulum which can become closed off to form a cyst (1,2). Other causes include cyst formation secondary to pericarditis, or a pericardial effusion causing increased intrapericardial pressure and herniation of the pericardial sac (1). While they can be found at either cardiophrenic angle or in the superior mediastinum, the right cardiophrenic angle is the most common (1,6). Discovery is often in a patient’s third or fourth decade of life, with no gender pre-disposition (2). If symptomatic, this is often from compressive symptoms of mass effect on surrounding structures including the heart, lungs, or esophagus. Cardiac compression can manifest as chest pain or congestive heart failure. Pulmonary or bronchial compression can present with chronic cough, dyspnea, or repeated respiratory infections. Esophageal compression can be associated with dysphagia (2,3). Rare, acute complications can include compression of the right mainstem bronchus, superior vena cava compression, cardiac tamponade, right ventricular outflow tract obstruction, cardiac arrythmias, or sudden death (1,4). There are few existing cases in the literature where hiccups were seen as a symptom of a pericardial cyst (7). Chest X-ray and CT are generally sufficient for diagnosis of a pericardial cyst. Treatment is typically conservative with radiographic follow-up (2). In symptomatic patients, or for growing cysts, surgical resection remains the treatment of choice. A VATS approach is preferred if possible.

In our case, hiccups were the sole symptom of presentation. Chest X-ray, CT, and MRI allowed for identification of the pericardial cyst during workup and for surgical planning. Interestingly, the hiccups were persistent but cyclical, without a clear inciting cause for this onset and offset. We hypothesize this could be due to pericardial fluid draining out of the pericardial cyst, giving symptomatic relief, before re-filling and compressing the right phrenic nerve causing hiccups to return. With the absence of hiccups after surgery and no return at 4 and 8 weeks post-operatively, it seems that the pericardial cyst was in fact the cause of this patient’s recurrent hiccups.

However, it is also possible that the anesthetic and neuromuscular blockade during this surgery may have contributed to outcomes. There are rare case reports showing general anesthesia with neuromuscular blockade and positive pressure ventilation as a method of treating persistent hiccups (10,11). It is unclear if this may have contributed to the patient’s outcome. Additionally, our case is limited to one patient. Further experience in elucidating hiccups as a symptom of pericardial cysts will help to clarify the incidence. This can therefore confirm both the utility of surgical resection when hiccups are a symptom, and of early chest imaging in the workup of recurrent hiccups.

Conclusions

In this case, we described a patient who presented primarily with recurrent hiccups, in which a pericardial cyst was identified and resected via a VATS approach, providing complete mitigation of symptoms. Pericardial cysts should be included in the differential diagnosis of recurrent hiccups, as surgical resection can be a curative treatment.

Acknowledgments

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://vats.amegroups.com/article/view/10.21037/vats-23-77/rc

Peer Review File: Available at https://vats.amegroups.com/article/view/10.21037/vats-23-77/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://vats.amegroups.com/article/view/10.21037/vats-23-77/coif). S.T. serves as an unpaid editorial board member of Video-Assisted Thoracic Surgery from April 2022 to December 2025 and reports financial relationships with Ethicon and Astra Zeneca. The other authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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