Pleuropericarditis complicated by a pericardial cyst

Abstract

We report the case of a 36-year-old woman with a recent upper respiratory illness who presented with chest pressure, lasting for five days, which improved with leaning forward. Physical examination and laboratory studies were unremarkable. Chest computed tomography scan revealed a 6.9 cm × 4 cm × 2.5 cm pericardial cyst. The patient was diagnosed with pleuropericarditis complicated by a pericardial cyst. Pericardial cysts are rare mediastinal lesions that are often congenital, but may be inflammatory. If symptomatic, surgical resection or percutaneous aspiration may be considered. In our patient, treatment with ibuprofen and colchicine resulted in a decrease in cyst size, suggesting an inflammatory component. We describe an uncommon case of reduction of a pericardial cyst shortly after treatment of pericarditis due to a probable viral infection. Non-operative management of pericardial cysts may be appropriate in these select cases.

<Learning objective: Pericardial cysts are rare, congenital, mediastinal lesions. Patients are often asymptomatic or may present with atypical chest pain, cough, or compressive symptoms. In previously reported cases, patients with pericardial cysts had undergone surgical intervention. Pericardial cysts may have an inflammatory component, and can be complicated by infections. Thus, non-operative management may be more appropriate in certain situations.>

Introduction

Pericardial cysts are rare, benign, mediastinal lesions with an estimated incidence of 1:100,000 [1]. They account for about 5–7% of all mediastinal tumors. The etiology is often congenital abnormal development of mesenchymal tissue, which results in a portion of the pericardium that is separated from the pericardial sac [2]. Although often congenital, pericardial cysts may be inflammatory in a few cases. If symptomatic, surgical resection or percutaneous aspiration may be considered. In most previously reported cases, patients with pericardial cysts had undergone surgical intervention. We describe a rare case of reduction of a pericardial cyst shortly after the treatment of pericarditis due to a probable viral infection.

Case report

Our patient is a 36-year-old woman with no significant past medical history who presented with chest pressure for five days. The pain was substernal in location, severe, and sharp in nature with no associated palpitations. Her symptoms worsened with inspiration and were relieved with leaning forward. She reported an upper respiratory illness about two weeks prior to presentation. She denied dyspnea, any history of trauma, dizziness, headache, or symptoms with exertion. She denied tobacco or illicit drug use. Her only medication was a combination oral contraceptive pill.

The patient was evaluated at an outside hospital with a chest radiograph, d-dimer, electrocardiogram, and laboratory tests, which were within normal limits. She returned to our facility with worsening chest pressure. On examination, temperature was 98 °F, blood pressure 130/70 mmHg, respiratory rate was 14, heart rate was 70 beats per minute, and oxygen saturation was 98% on ambient air. Cardiopulmonary examination was unremarkable. Laboratory data showed a normal blood count, kidney and liver function, and no electrolyte abnormalities. Erythrocyte sedimentation rate (ESR) was elevated at 42 mm/h and C-reactive protein (CRP) was found to be 5 mg/L. Cardiac enzymes were within normal limits. A pregnancy test was negative. Electrocardiogram showed no evidence of ischemia. Transthoracic echocardiogram (TTE) demonstrated a normal left ventricular ejection fraction and a 4 cm mass adjacent to the right atrium. The patient underwent a computed tomography (CT) scan of the chest, and cardiac magnetic resonance (CMR) imaging, which revealed a circumscribed cystic mass 6.9 cm × 4 cm × 2.5 cm adjacent to the right pericardial border, consistent with a pericardial cyst (Fig. 1, Fig. 2). She was diagnosed with pleuropericarditis complicated by a pericardial cyst. She received treatment with ibuprofen for seven days and colchicine for three months with subsequent improvement in symptoms over several weeks. Follow-up TTE showed a decrease in cyst size after treatment (Fig. 2). Inflammatory markers, including ESR and CRP, had trended down to 7 mm/h and 1 mg/L, respectively.

Fig. 1.

Coronal views, computed tomography scan (a), cardiac magnetic resonance (CMR), steady-state free precession image (b), and CMR T2 half-Fourier acquisition single-shot turbo spin echo image (c). RA, right atrium; RV, right ventricle; LV, left ventricle; IVC, inferior vena cava; SVC, superior vena cava; AA, ascending aorta; DA, descending aorta.

Fig. 2.

Axial views, cardiac magnetic resonance (CMR) images. Top row: Steady-state free precession image (a), volumetric interpolated breath-hold examination image (b), T2 half-Fourier acquisition single-shot turbo spin echo image (c). Bottom row: CMR late gadolinium enhancement images revealing no enhancement (d), CMR, phase sensitive inversion recovery image (e). RV, right ventricle; LV, left ventricle; IVC, inferior vena cava; DA, descending aorta.

Discussion

Patients with pericardial cysts are usually asymptomatic. About one third of the patients may present with atypical chest pain, dyspnea, or persistent cough [3]. The most common site of involvement is the right cardiophrenic angle (51–70%), followed by left cardiophrenic angle (22–38%), or superior part of the mediastinum (8–11%) [2]. TTE can delineate the margins and cystic characteristics of the echolucent mass [3]. Contrast chest CT scan is the imaging modality of choice for diagnosing pericardial cysts. The cysts appear as thin-walled, sharply demarcated, oval masses with an attenuation of 30–40 Hounsfield units that do not enhance with intravenous contrast [4]. On CMR imaging, these cysts may appear as low to intermediate intensity on T1-weighted images and high signal intensity on T2-weighted sequences that do not enhance with contrast [4]. Their size ranges from 2 cm to 28 cm. Our patient was found to have a cystic mass 6.9 cm × 4 cm × 2.5 cm, adjacent to the right pericardial border (Fig. 1, Fig. 2).

Management depends on the presence of symptoms and cyst characteristics. Asymptomatic patients are treated conservatively and undergo serial TTE to monitor cyst size [3]. Percutaneous drainage or aspiration of the cyst may be performed, but this approach has a 30% recurrence rate. Resection is considered when the diagnosis is uncertain, or symptoms are persistent, or if the cyst has potential to be malignant, or when complications have occurred [1], [5]. Video-assisted thorascopic surgery and robotic resection are the most common methods of resection [3].

Our patient presented with pleuropericarditis and was found to have a pericardial cyst. Treatment with ibuprofen and colchicine led to a reduction in cyst size. This suggests an inflammatory component of the cyst, however an underlying congenital etiology cannot be entirely excluded. It is unclear whether an underlying cyst was exacerbated by pericarditis or predisposed the patient to developing pericarditis or if pericarditis led to the formation of an inflammatory cyst. Inflammatory cysts, pseudocysts often associated with pericardial effusions, may occur due to rheumatic heart disease, infection such as tuberculosis, trauma, and cardiac surgeries [5]. Familial Mediterranean fever, an autosomal recessive disorder mainly affecting Turks, Armenians, Arabs, and Jews, manifests as recurrent fever and inflammation of the peritoneal, synovial, and pleural surfaces, but can also rarely present with pericarditis and inflammatory cysts [6], [7].

Our patient likely had an inflammatory cyst related to a viral syndrome as she reported an upper respiratory illness prior to her symptoms. Previously, spontaneous reduction in cyst size has been noted at 10-month follow-up [8]. Other reported cases of resolution of pericardial cysts involved patients who had undergone invasive procedures [9], [10]. However, we describe a rare case of reduction of a pericardial cyst shortly after treatment of pericarditis due to a probable viral infection. In previous cases, patients had surgical excision to avoid further complications. Our case illustrates that perhaps a more conservative approach may be taken for certain patients with pericardial cysts with an inflammatory component. Non-operative management may be appropriate for these select patients.

Conflict of interest

The authors declare no conflict of interest. No funding was received.

Appendix A. Supplementary data

The following are the supplementary data to this article:

Video 1

Transthoracic echocardiography subcostal view revealing an echolucent mass that measures approximately 6.9 cm × 4 cm prior to treatment.

Video 2

Repeat transthoracic echocardiography revealing the mass, now decreased in size to about 3.5 cm × 1.5 cm, post treatment.