SESSION TITLE: Interstitial Lung Disease Global Case Reports
SESSION TYPE: Global Case Report
PRESENTED ON: Tuesday, October 29, 2013 at 01:30 PM - 02:30 PM
INTRODUCTION: Macrolide have been proven to be beneficial in the treatment of chronic respiratory inflammatory diseases. Here we report a case of desquamative interstitial pneumonia (DIP) with recurrent spontaneous pneumothorax , in which treatment with corticosteroids was unsuccessful. Clinical and radiologic symptoms improved with a combination therapy with clarithromycin and prednisolone; However, tapering the dose of prednisolone decreased efficacy of clarithromycin, resulting in an unfavorable response. This finding supports the efficacy of combination therapy with macrolide and corticosteroid for cases of DIP refractory to corticosteroids.
CASE PRESENTATION: A 34-year-old man was admitted with a 4-month history of dry cough and progressive dyspnea on exertion. He was a 15-pack-year smoker. In serologic tests, C-reactive protein, rheumatoid factor, antineuclear antiboby test were negative. Pulmonary function tests revealed a restrictive disease of moderate severity. A chest X-ray was normal; however, chest computed tomography (CT) scan showed diffuse ground-glass densities. Wedge resection via video-assisted thoracic surgery (VATS) was performed, and microscopic findings were mildly fibrotic and thickened alveolar septa with macrophages prominently accumulated in alveolar spaces, finally diagnosed as DIP. He was encouraged to quit smoking and started on prednisolone (1mg/kg/day) at discharge. The dose of prednisolone was tapered to 30 mg daily in outpatient clinic. Four months later, the patient returned complaining of sudden dyspnea and left chest wall pain. Chest radiograph revealed left tension pneumothorax, and tube thoracostomy was performed. However, the left lung failed to fully expand by the third hospital day; thus, wedge resection via VATS was performed. Moreover, right pneumothorax developed on the seventh hospital day, which failed to completely recover even after tube thoracostomy; therefore, right upper lobe lobectomy was performed. Histological analysis of the excised lung specimens was similar to the initial biopsy. The persistent ground-glass opacities on chest CT revealed that the patient was refractory to corticosteroid therapy. The patient was started the combination therapy with clarithromycin (500mg, twice daily) and prednisolone (30mg, daily). Within two weeks, the ground-glass opacities started to disappear on chest CT, and exertional dyspnea was getting improved. After 2 months of treatment, the patient reported considerable improvement in the symptoms. He continued with the combination therapy and the dose of prednisolone was tapered to 15 mg daily over two months. However, progressive dyspnea returned with no evidence of infection. Chest radiograph revealed moderate amount of left pneumothorax and tube thoracostomy was performed. His symptoms improved upon treatment with an increased dose of prednisolone with clarithromycin. The patient has maintained this improved state; and has been receiving prednisolone (20mg daily) and clarithromycin (500mg twice daily) in an outpatient clinic for 2 months.
DISCUSSION: The presented case report supports the combination therapy with macrolide and corticosteroid based on clinical and radiological findings. The use of macrolide antibiotic is supported by a recent case reporting the beneficial effect of clarithromycin in a patient with DIP refractory to corticosteroids. The presented report clearly shows that macrolide efficacy requires co-administration of corticosteroids.
CONCLUSIONS: This report proposes the combination therapy with macrolide and corticosteroid for cases of DIP refractory to corticosteroid monotherapy.
Reference #1: Hartman TE, Primack SL, Kang EY, et al. Disease progression in usual interstitial pneumonia compared with desquamative interstitial pneumonia. Assessment with serial CT. Chest 1996; 110:378-382
Reference #2: Ryu JH, Myers JL, Capizzi SA, et al. Desquamative interstitial pneumonia and respiratory bronchiolitis-associated interstitial lung disease. Chest 2005; 127:178-184
Reference #3: Knyazhitskiy A, Masson RG, Corkey R, et al. Beneficial response to macrolide antibiotic in a patient with desquamative interstitial pneumonia refractory to corticosteroid therapy. Chest 2008; 134:185-187
DISCLOSURE: The following authors have nothing to disclose: Dong Won Park, Hyun Jung Kwak, Ji-Yong Moon, Sang-Heon Kim, Tae-Hyung Kim, Jang Won Sohn, Dong Ho Shin, Ho Joo Yoon, Sung Soo Park
No Product/Research Disclosure Information