A CURIOUS MANIFESTATION OF SARCOIDOSIS

Abstract

SESSION TITLE: Pulmonary Manifestations of Systemic Disease SESSION TYPE: Fellow Case Reports PRESENTED ON: 10/21/2019 3:15 PM - 4:15 PM INTRODUCTION: Patients with sarcoidosis have pulmonary involvement greater than 90% of the time, however that typically involves intra-thoracic lymph nodes and lung parenchyma, with pleural involvement being exceedingly rare.[1,2,3] We present a case of a new onset unilateral pleural effusion in a patient with sarcoidosis. CASE PRESENTATION: A 68 year old female with a biopsy proven diagnosis of pulmonary sarcoidosis presented to the pulmonary clinic with complaints of a non-productive cough and worsening dyspnea on exertion. She noted that over the previous four months she had a decreased exercise capacity when running on the treadmill. She trialed discontinuing her beta-blocker, however, her exertional dyspnea symptoms persisted. Chest radiography revealed a new right-sided moderate sized effusion. Pleural fluid analysis showed a lymphocyte predominant exudative effusion while cytology and cytometry were negative for malignancy. Due to her recurrent effusion she underwent pleuroscopy for pleural biopsy with indwelling pleural catheter placement. Parietal pleura biopsies showed mesothelial cell proliferation concerning for mesothelioma, however BAP-1 immunostaining was negative along with no evidence of homozygous p16 deletion on fluorescence in situ hybridization. As the histologic workup was non-conclusive the patient underwent video-assisted thoracoscopic surgery. Right middle and lower lobe wedge resection showed non-caseating granulomas and were negative for acid-fast bacilli and fungal stains. Subsequently due to her symptomatic recurrent pleural effusion she was started on steroid therapy with improvement of parenchymal lesions, lymphadenopathy, and pleural effusion noted on imaging within three weeks of initiation. DISCUSSION: Pleural effusions related to sarcoidosis have been described in the medical literature predominantly as case reports/series. A recent prospective study using pleural ultrasound to identify pleural effusions in patients with known sarcoidosis noted an incidence of only 1.1%.[2] Pleural effusions are typically noted to be lymphocyte predominant exudates, however, transudates, chylothorax, and hemothorax have all been described. The pleural fluid to serum protein ratio, compared to the LDH criteria, is typically noted to be in the exudative range.[2] Due to the variability and non-specific findings on pleural fluid analysis, diagnosis should be guided by biopsy confirmation. Treatment should be reserved for patients with recurrent effusions, which are typically responsive to steroid therapy. CONCLUSIONS: As a result of the relative rare nature of pleural effusions in patients with sarcoidosis, it is imperative to confirm the true etiology of the effusion. Although pleural fluid analysis is useful, pleural biopsy remains the gold standard diagnostic test in the workup of an undiagnosed pleural effusion and should be utilized in the workup of such in patients with sarcoidosis. Reference #1: Sharma OP, Gordonoson J. Pleural effusion in sarcoidosis: a report of six cases. Thorax. 1975; 30:95–101 Reference #2: Huggins JT, Doelken P, Sahn SA, King L, Judson MA. Pleural effusion in a series of 181 outpatients with sarcoidosis. Chest. 2006; 129:1599–1604 Reference #3: Baughman RP, Teirstein AS, Judson MA, et al. Clinical characteristics of patients in a case control study of sarcoidosis. Am J Respir Crit Care Med. 2001; 164:1885–1889 DISCLOSURES: No relevant relationships by Sara Shadchehr, source=Web Response No relevant relationships by Dhaval Thakkar, source=Web Response