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


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

Vasileios Karamouzos et al. J Investig Med High Impact Case Rep.


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.

Keywords: Langerhans cell; bilateral; histiocytosis; interstitial; lung diseases; pneumothorax; pulmonary.

Conflict of interest statement

Declaration of Conflicting Interests: The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.


Figure 1.
Figure 1.
Chest radiograph with bilateral pneumothorax at presentation.
Figure 2.
Figure 2.
Bilateral lung re-expansion after bilateral drainage tube insertion.
Figure 3.
Figure 3.
Chest computed tomography scan showing multiple, small, thin-walled, well-defined, rounded cysts that were evenly distributed throughout both lungs.
Figure 4.
Figure 4.
Pulmonary Langerhans cell histiocytosis pathophysiology. BRAF; MAP2K1, mitogen-activated protein 2 kinase 1; MAP3K1, mitogen-activated protein 3 kinase 1; TNFa, tumor necrosis factor alpha; GM-CSF, granulocyte-macrophage colony-stimulating factor; TGF-beta, transforming growth factor beta; CCL20, chemokine (C-C motif) ligand 20.

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