Simultaneous Bilateral Spontaneous Pneumothorax in an Adult Patient With Pulmonary Langerhans Cell Histiocytosis: A Case Report.

Vasileios Karamouzos,Dimitrios Velissaris,Christina Kalogeropoulou,Nikolaos Panagiotopoulos,Evangelos Karanikolas,Christos Prokakis,Efstratios Koletsis,Fotini Kosmopoulou,Diamanto Aretha

doi:10.1177/2324709618792945

Abstract

We report a case of a young female with known history of pulmonary Langerhans cell histiocytosis who was initially presented in the emergency department of a university hospital with respiratory distress. Clinical assessment and diagnostic workup revealed left hemithorax subcutaneous emphysema, bilateral pneumothorax, and atelectasis in both lower lung lobes. The patient was treated with bilateral staged thoracoscopic bullectomy and mechanical abrasion of the parietal pleura combined with chemical pleurodesis with talc. A new occurrence of right-sided pneumothorax was noticed 3 days after surgery, which was treated with chest tube insertion and chemical pleurodesis. The aforementioned surgical approach resulted in complete lung expansion and the patient’s full recovery. A review of pulmonary Langerhans cell histiocytosis and treatment options in cases of pneumothorax due to lung histiocytosis is also presented in this report.

Highlights

Langerhans cell histiocytosis (LCH) belongs to a group of rare histiocytic disorders characterized by organ infiltration of Langerhans cells causing inflammation and tissue damage
Respiratory distress due to pneumothorax is a common presentation in the emergency department (ED), clinicians should keep in mind that one presented with spontaneous pneumothorax may conceal an uncommon disease
We report a case of a 20-year-old woman suffering from pulmonary LCH (PLCH) with simultaneous bilateral pneumothorax, treated initially with thoracoscopic bullectomy combined with mechanical abrasion of the parietal pleura and chemical pleurodesis

Summary

Introduction

Langerhans cell histiocytosis (LCH) belongs to a group of rare histiocytic disorders characterized by organ infiltration of Langerhans cells causing inflammation and tissue damage. We report a case of a 20-year-old woman suffering from PLCH with simultaneous bilateral pneumothorax, treated initially with thoracoscopic bullectomy combined with mechanical abrasion of the parietal pleura and chemical pleurodesis. Chest auscultation revealed absence of air entry bilaterally in the upper-mid zones and substantially reduced in both the lower zones anteriorly and posteriorly. She was treated with paracetamol (500 mg tds [3 times daily] intravenously [IV]), ampicillin/sulbactam (3 g qds [4 times daily] IV), ranitidine (150 mg bd [twice daily] IV), and levothyroxine sodium (50 μg od [once daily] po [orally]). The patient’s hemodynamic and respiratory status remained stable, laboratory testing did not reveal any abnormalities, and she was discharged to home 4 days later, completely asymptomatic, with radiologic confirmation of complete lung expansion

Findings

Discussion

Conclusion