Intrapleural Corticosteroid Injection in Eosinophilic Pleural Effusion Associated with Undifferentiated Connective Tissue Disease
A 67-year-old man was admitted with a 1-month history of increasing dyspnea of modified Medical Research Council grade 3. The patient had undergone mitral valve replacement 20 years earlier and had been treated for heart failure, diabetes, and hypertension. His medications included warfarin, digoxin, furosemide, losartan, isosorbide mononitrate, glimepiride, and thiotacid. He had no smoking history. At the time of hospitalization, the patient was alert and the following findings were noted in a physical examination: blood pressure, 140/60 mm Hg; pulse rate, 84 beats/min; respiratory rate, 20 breaths/min; and body temperature, 37.6℃. He had no history of chest trauma or any prominent abnormalities on chest, abdominal, and lower-extremity examinations. Other results of the physical examination were non-specific.\nOn a physical examination, decreased breath sounds were auscultated on the left lower thorax. A chest radiograph showed blunting of the left costophrenic angle (). Chest computed tomography images obtained after therapeutic thoracentesis revealed pleural thickening and calcification on the right posterior pleura but no nodule, mass, or lesion in the lung parenchyma showing in connective tissue disease (). Transthoracic echocardiography revealed enlargement of both atria, dilatation of the left-sided ventricle, and concentric left ventricular hypertrophy with left ventricle ejection fraction 57%. Mild mitral regurgitation and tricuspid regurgitation (grade II) were noted, but no definite change could be observed. There was no pericardial effusion or pericardial thickening, and no valvular vegetations were seen. Although a dose of diuretics had been added for several days, the amount of the left pleural effusion increased markedly, and the patient was referred to a pulmonologist. Laboratory examinations showed the following findings: white blood cell count, 9,300/mm3; hemoglobin, 11.7 g/dL; platelet count, 338,000/mm3; aspartate aminotransferase, 29 IU/L; alanine aminotransferase, 24 IU/L; total protein, 7.8 g/dL; blood urea nitrogen, 22 mg/dL; creatinine, 1.4 mg/dL; C-reactive protein, 8.3 mg/dL; and NT-pro B-type natriuretic peptide (BNP), 1,324 pg/mL. Arterial blood gas analysis in room air yielded results within normal ranges. Analysis of the pleural effusion revealed the following findings: color, yellow; pH, 7.4; specific gravity, 1.010; red blood cell count, 13,760/mm3; white blood cell count, 990/mm3 (neutrophils, 2%; lymphocytes, 29%; eosinophils, 34%); protein, 4.2 g/dL; lactate dehydrogenase, 384 U/L; glucose, 117 mg/dL; carcinoembryogenic antigen, 0.2 ng/mL; adenosine deaminase, 31 IU/L; negative acid-fast staining, culture, non-specific cytology, and negative results in real-time polymerase chain reaction for Mycobacterium tuberculosis. Diagnostic thoracentesis revealed a pleural effusion appearing as an exudate with an elevated eosinophil count (34%). Serological tests for parasites (Clonorchis sinesis, Paragonimus westermani, Cysticercus cellulosae, Sparganum) yielded negative results. Serological test for rheumatologic diseases and vasculitides yielded positive results for the antinuclear antibody (>1:1,280, speckled pattern) and the anti-SS-A/Ro (179.09 EU) and anti-SS-B/La antibodies (208.98 EU). Serological tests for rheumatoid factor (RF) (<20 IU/mL), anti-ds DNA, and complement (C3, C4) yielded negative results. Of the medications the patient was receiving, although warfarin was associated with EPE, he had been taking the drug without any side effects since the mitral valve replacement surgery. Therefore, we considered that the possibility of warfarin-associated EPE was low. The patient complained of xerostomia but not of xerophthalmia, Raynaud's phenomenon, skin lesion, or arthralgia. The patient's heart failure was well controlled; he showed no pitting edema, and his NT-pro BNP level had reduced from 2,419 pg/mL to 1,324 pg/mL in a 1-month period. EPE associated with UCTD was considered, and the patient was discharged after 1 L of excess fluid was removed by therapeutic thoracentesis. About 2 weeks later, his dyspnea worsened and the left pleural effusion increased again. Repeated therapeutic thoracentesis temporarily relieved the symptoms, but they aggravated again within 2 weeks. The administration of systemic corticosteroid was considered but was contraindicated by chronic anticoagulation therapy, uncontrolled blood sugar levels, and the patient's age.\nBecause we were reluctant to administer systemic corticosteroid therapy, a single dose of 125 mg methylprednisolone was injected directly into the pleural space. A chest X-ray image obtained 2 weeks later showed a marked decrease in the pleural effusion (). No recurrence was observed during a 1-year follow-up, and serological tests for autoantibodies yielded similar results to those obtained in the previous year ().