Fatal congenital lobar emphysema in a puerpera: a case report and literature review

Background Congenital lobal emphysema (CLE) is a developmental lung abnormality usually diagnosed in the neonatal period and is rarely observed in adults. Adults with CLE are usually asymptomatic and only a small fraction may present with coughing, recurrent pneumonia and respiratory distress. In imaging studies, the most frequently affected lobe of CLE is the left upper lobe, followed by the right middle lobe. However, multilobar involvement with severe mediastinal shift is extremely rare. Case presentation We report a case of fatal CLE in a 28-year-old puerpera with postpartum respiratory failure. Chest computed tomography (CT) revealed emphysema of the right upper, middle and lower lobes resulting in adjacent atelectasis. Hyperinflation of the right upper lobe crossed the midline, leading to a deviation of the mediastinal structure to the left hemithorax and severe compression of the left lung. Conclusions Early and timely diagnosis of CLE with routine follow-up is necessary for patients. CLE, especially with multilobar involvement or mediastinal shift, could be life-threatening and should be promptly and aggressively treated to prevent severe complications.

Diffuse Idiopathic Neuroendocrine Cell Hyperplasia (DIPNECH) on Electromagnetic Navigation Bronchoscopy (EMN) Guided Fine Needle Aspiration (FNA): A Case Report

Introduction/Objective DIPNECH is a clinicopathological diagnosis characterized by abnormal proliferation of single or clusters of neuroendocrine cells in the bronchial mucosa. The World health organization includes DIPNECH as a preinvasive lesion to carcinoid tumors of the lung. Diagnosis is often delayed or missed due to insidious presentation and rarity of the disease. High degree of suspicion in the appropriate clinical and radiological context is important for early diagnosis. We describe a unique case of DIPNECH diagnosed by EMN guided aspiration cytology in the setting of multiple incidental lung nodules and confirmed on surgical resection. Methods/Case Report A 69-year-old female with history of insidious cough was referred to pulmonology clinic for multiple incidental bilateral lung nodules detected by CT scan, largest involving the right middle lobe. Although the main diagnostic consideration was pulmonary metastasis, several radiological features, including diffuse mosaic attenuation pattern, prompted the possibility of a diffuse neuroendocrine process. Electromagnetic navigation bronchoscopy (ENB) guided fine needle aspiration cytology was performed. Air-dried and Papanicolaou stained smears were scant but showed dispersed and clusters of small cells with fine chromatin with minimal cytoplasm. No necrosis or mitosis were seen. A cell block was prepared that showed rare clusters of tumor cells positive for immunostains synaptophysin, chromogranin, CD56, with a Ki-67 of 1%. In the appropriate clinical and radiological context findings were consistent with a well differentiated neuroendocrine process such as DIPNECH. Right middle lobe resection confirmed extensive DIPNECH with multiple typical carcinoid tumors and tumorlets. Results (if a Case Study enter NA) NA Conclusion It is essential to be familiar with the clinical and radiological findings of DIPNECH and include it in the differential diagnosis of its mimickers. Although the diagnosis is challenging on limited specimens, this report indicates that EMN guided aspiration cytology can be used as a valuable early tool for accurate diagnosis and timely management of DIPNECH.

Case report: pulmonary nocardiosis caused by Nocardia exalbida in an immunocompetent patient

Background Nocardiosis is known as an opportunistic infection in immunocompromised hosts, but it occasionally has been reported in immunocompetent patient. The Nocardia exalbida is first-reported in 2006 from Japan, and a few cases of have been reported in only immunocompromised host, and the characteristic is still unclear. We herein describe the first case of pulmonary nocardiosis caused by N. exalbida in an immunocompetent patient. Case presentation A77 -year-old Japanese man was admitted to our hospital on November 2, 2018. He was a lifelong non-smoker with no childhood history of respiratory disease. He had a medical history of dyslipidemia. One month before this admission fevers, sputum, mild cough were developed and he was evaluated in a clinic near our hospital. His diagnosis was community acquired pneumonia within his right middle lobe. He was treated with ceftriaxone 1 g/day intravenously for a week, however his symptoms relapsed a few days later. So, the physician retried ceftriaxone for another 3 days, but his symptoms did not improve. He was referred to our hospital. He was treated with sitafloxacin as an outpatient for a week, however his symptoms got worse. The chest CT showed consolidation and atelectasis in his right middle lobe. Low density area was scattered in consolidation, and right pleural effusion was observed. The patient was diagnosed with pulmonary abscess and he was admitted. Administration of piperacillin/tazobactam improved his condition. We switched antibiotics to amoxicillin/clavulanate, and he was discharged. After 2 weeks, he relapsed and was admitted again. After administration of piperacillin/tazobactam for 3 weeks, we perform bronchoscopy and Nocardia species were cultured from samples of the bronchial wash. The isolates were identified as N. exalbida using 16S rRNA gene sequencing. We prescribed Trimethoprim / Sulfamethoxazole (TMP/SMX) for 4 months. Then we switched to minocycline for renal dysfunction caused from TMP-SMX for 1 more month. After 5 months therapy, Consolidation on CT disappeared, and Nocardiosis was cured. Conclusion we reported the first case of pulmonary nocardiosis caused by N. exalbida in an immunocompetent patient. N. exalbida infection might be associated with a good response to treatment.

Unusual case of chronic cavitary pulmonary aspergillosis presenting as spontaneous pneumothorax in an immunocompromised man

Chronic cavitary pulmonary aspergillosis (CCPA) is a slow destructive type of chronic pulmonary aspergillosis, characterised by multiple pulmonary cavities that develop and expand over several months or years. Pleural involvement in the form of pneumothorax has been rarely reported in CCPA. We report such an unusual case of an immunocompromised male, with a history of chronic cough and fever, presenting with acute onset of shortness of breath. Chest imaging showed bilateral multiple cavitating nodules, ground glass opacities and dense right middle lobe consolidations and right-sided pneumothorax. Bronchoalveolar lavage (BAL) showed septate hyaline hyphae on KOH (potassium hydroxide) staining and fungal culture grew Aspergillus fumigatus. BAL and serum galactomanan were positive and serum IgG for A. fumigatus was 58 MgA/L (0–40MgA/L) confirming the cause of spontaneous secondary pneumothorax in our patient as CCPA.