A Case Report of Hemophagocytic Lymphohistiocytosis and Meningitis Due to Atezolizumab Treatment for Lung Adenocarcinoma.

Immune checkpoint inhibitors (ICIs) are used to treat a variety of tumors. Despite their broad beneficial effects, these inhibitors can cause immune-related adverse events (irAEs) and even death. Hemophagocytic lymphohistiocytosis (HLH) and meningitis, although infrequent, can be aggressive and life-threatening due to excessive immune activation. Herein, we report a case of an 80-year-old man who developed HLH after receiving atezolizumab monotherapy as a second-line treatment for lung adenocarcinoma. He was treated for HLH with oral prednisolone (PSL), but further ataxia and dysuria developed, and a lumbar puncture diagnosed meningitis. Both HLH and meningitis improved with continued oral PSL treatment. This is the first case of atezolizumab-induced HLH with meningitis and highlights the importance of early diagnosis and treatment for rare irAE.

The prevalence of obstructive sleep apnea in Japanese asthma patients.

BACKGROUND: Obstructive sleep apnea (OSA) occurs more commonly in asthma patients than in the general population because these conditions share some comorbidities. In Japan, the prevalence of OSA in the general population is reported to be approximately 20%; however, few reports have described the prevalence of OSA in asthma patients. Furthermore, the characteristics of Japanese patients with OSA and asthma are not clear. METHODS: Adult asthma patients were recruited from the outpatient departments of our institution between August 31, 2017, and March 31, 2019. In all included patients, the presence and severity of OSA were evaluated by the Epworth Sleepiness Scale (ESS) and a home sleep test (HST) using portable polysomnography (PSG). The rate of coexisting OSA in asthma patients and the characteristics of those patients according to the severity of OSA were investigated. RESULTS: Fifty-three patients were included. OSA was detected in 36 (67.9%) patients (mild, n = 15; moderate, n = 14; and severe, n = 7). Patients with OSA had significantly higher body mass index, Brinkman index, apnea-hypopnea index (AHI), and 3% oxygen desaturation index (ODI) values in comparison to those without OSA, while the percentage of the predicted value of forced vital capacity (%FVC) and lowest SpO2 levels were significantly lower. As the severity of OSA increased, age, brain natriuretic peptide level, AHI, and 3%ODI increased, and in contrast, FVC, %FVC, forced expiratory volume in one second (FEV1), percentage of the predicted value of FEV1 (%FEV1), Epworth Sleepiness Scale (ESS), 3%ODI, and lowest SpO2 levels decreased. In particular, the fact that the ESS value was inversely correlated with the severity of OSA in our patients was different from the general characteristics of OSA. Moreover, the AHI value was negatively correlated with FVC, %FVC, FEV1, and %FEV1. BMI was the only independent factor for the presence of OSA, and for asthma severity (FEV1, % of predicted), there was a weak correlation with smoking history. CONCLUSIONS: This is the first report to investigate the prevalence of OSA in Japanese asthma patients, using an HST. This study suggests that an HST should be performed in addition to the sleep interview for asthma patients with refractory disease, a low pulmonary function, advanced age, and high BMI because the more severe the OSA, the lower the ESS value may be.

Osimertinib for Lung Squamous Cell Carcinoma: A Case Report and Literature Review.

The efficacy of epidermal growth factor receptor tyrosine kinase inhibitors (EGFR-TKIs) in lung squamous cell carcinoma is said to be low. Thus far, only four cases of osimertinib in lung squamous cell carcinoma have been published. We experienced a case of EGFR mutant lung squamous cell carcinoma in which fifth-line treatment with osimertinib was effective after T790M EGFR mutation turned positive. Osimertinib was resumed after sixth-line chemotherapy was ineffective, showing efficacy again. Osimertinib may be a promising treatment option for EGFR mutant lung squamous cell carcinoma. This is the first report to show its effect in a case of rechallenge after intervening chemotherapy. It may therefore be important to evaluate EGFR in never-smoker lung squamous cell carcinoma patients.

A familial case of Birt-Hogg-Dubé syndrome complicated with various cancers.

An 89-year-old woman with small papules on her face presented to our hospital complaining of progressive dyspnoea. Chest computed tomography (CT) showed bilateral multiple lung cysts, a nodular opacity in the right lower lobe, and bilateral pleural effusion. She was diagnosed with adenocarcinoma. Her son, a 65-year-old man, also had bilateral basally located lung cysts and a past medical history of spontaneous pneumothorax. He had multiple papules on the face and neck, which were pathologically diagnosed as fibrofolliculomas. We considered these cases to be Birt-Hogg-Dubé syndrome (BHDS). Folliculin (FLCN) gene mutations that may be tumour suppressive are suspected to be causative of this syndrome. FLCN dysfunction might lead to the development of various types of tumours other than renal tumours.

Primary Pleural Melanoma: A Case Report and Literature Review.

Primary pleural melanoma is an extremely rare neoplasm, and to the best of our knowledge, there have been only 8 case reports of this condition in the English literature. We herein report a rare case in which the cytological and immunocytochemical analyses of pleural fluid and ultrasonography (US)-guided biopsy of a pleural lesion were useful for the diagnosis primary pleural melanoma. This case highlights the importance of careful physical examinations, cytomorphologic and immunocytochemical analyses of pleural fluid, as well as the utility of US-guided biopsy of the pleural lesions in the diagnosis of primary pleural melanoma.

Pneumocystis jirovecii Pneumonia in an Immunocompetent Japanese Man: A Case Report and Literature Review.

We herein report the case of a 37-year-old immunocompetent man who died from Pneumocystis jirovecii pneumonia (PCP). He was initially treated for an acute exacerbation of interstitial pneumonia; however, the elevation of the patient's serum (1-3) ß-D glucan (BG) level suggested the possibility of PCP and sulfamethoxazole trimethoprim was added. A postmortem pathological examination and retrospective Grocott's methenamine silver (GMS) staining of the bronchoalveolar lavage fluid (BALF), which was obtained on the day of admission, revealed PCP. The present case suggests that it is essential to perform a BG assay and GMS staining of BALF specimens when patients show diffuse ground-glass opacity on chest computed tomography, regardless of their immune status.

Repetitive saliva swallowing test and water swallowing test may identify a COPD phenotype at high risk of exacerbation.

INTRODUCTION: Patients with chronic obstructive pulmonary disease (COPD) are known to present with dysphagia from an early stage. Dysphagia leads to swallowing-related complications, in turn leading to COPD exacerbation. Dysphagia screening is recommended; however little is known of its utility in detecting a COPD phenotype at risk for exacerbation. The simple swallowing provocation test (SSPT), considered to be a standard screening test, requires specific equipment, physician skill and patient discomfort. OBJECTIVES: The aim of this study was to find an easier and less invasive measure to screen for dysphagia in patients with COPD. METHODS: We retrospectively reviewed patients with COPD who were screened for dysphagia [Repetitive saliva swallowing test (RSST), water swallowing test (WST), SSPT and a questionnaire] from June to November 2016. The patients were classified into two groups according to the presence of COPD exacerbation in the past 3 years (ie, exacerbation and non-exacerbation group), and the dysphagia screening results were compared between the groups. RESULTS: Of the 80 cases included, 42 had 1 or more exacerbations in the past 3 years (exacerbation group), and 38 had none (non-exacerbation group). Statistically significant differences between the groups were observed in the RSST, and vocal change in the WST (P < 0.05). There were no significant differences between the groups in the results of SSPT, COPD stage and other clinical status. CONCLUSIONS: Compared to the SSPT, RSST and WST may be more appropriate screening methods in patients with COPD. A prospective study is necessary for further assessment.

A case of huge lymphatic and venous malformations of the mediastinum.

We herein report a case of huge lymphatic and venous malformations of the mediastinum (LVM). A 19-year-old man presented for evaluation of a mediastinal mass. On chest computed tomography, the mass demonstrated contrast enhancement and showed dilated veins draining into the superior and inferior vena cava, with multiple scattered calcifications. The lesion was enhanced heterogeneously on T1-weighted magnetic resonance imaging (MRI) and hyperintense on T2-weighted MRI. Contrast-enhanced MRI revealed that the mass was enhanced, with the multilocular part marginally enhanced. From these images, we diagnosed him with LVM. Given that an operation presented a high risk, we decided to follow him up without any treatment.

"Pulmonary Actinomycosis attributable to Actinomyces meyeri presenting as cardiac tamponade: a case report".

BACKGROUND: Recently, it is reported that Actinomyces meyeri is the most common species causing actinomycosis. However, to our knowledge, there was no report about pulmonary actinomycosis attributable to A. meyeri presenting as cardiac tamponade. CASE PRESENTATION: Hereby we describe a case of pulmonary actinomycosis attributable to A. meyeri presenting as cardiac tamponade. At first, the patient was diagnosed with bacterial pericarditis with lung abscess in the left lower lung lobe and underwent pericardial drainage. Three days after the removal of the drainage tube, atrial fibrillation followed by cardiac arrest with asystole occurred and immediate cardiopulmonary resuscitation restored his circulation. Thereafter, he underwent pneumocentesis of the lung abscess and the culture grew A. meyeri. He was successfully treated with penicillin G. CONCLUSION: This is the first case of pulmonary actinomycosis attributable to A. meyeri presenting as cardiac tamponade. We believe that an increased awareness of the disease is necessary to expedite diagnosis therefore minimizing morbidity and mortality.

Peripheral T cell lymphoma not otherwise specified (PTCL-NOS) presenting as an endobronchial lesion: Case report and literature review.

Peripheral T cell lymphoma not otherwise specified (PTCL-NOS) is a rare entity of lymphoma. We herein report an even rarer case of a 68-year-old male with PTCL-NOS presenting as an endobronchial lesion, and review previously published cases in the literature. Initially, he was referred to our hospital for further investigation of the right upper lobe consolidation on chest radiograph. Computed tomography and 18F-fludeoxyglucose positron emission tomography revealed a right hilar mass with obstruction of the main bronchus and submandibular, right axillary and mediastinal lymphadenopathy. Pathological examination of the biopsy specimens from of the endobronchial lesion and subcutaneous nodule revealed PTCL-NOS. Chemotherapy was started but he finally died due to septic shock after the second-line chemotherapy.

Miliary Tuberculosis in a Young Woman with Hemophagocytic Syndrome: A Case Report and Literature Review.

We herein report a rare case of miliary tuberculosis-associated hemophagocytic syndrome (HPS) complicated with respiratory failure. A 19-year-old Japanese woman with a fever, general malaise, and chest radiograph abnormalities was referred to our hospital. After admission, she developed respiratory failure with pancytopenia. A histological examination of lung and bone marrow biopsy samples revealed noncaseating granulomas without evidence of acid-fast bacilli or lymphoma. In addition, a bone marrow biopsy showed marked histiocyte hyperplasia with hemophagocytosis, and a bronchoalveolar lavage fluid culture grew Mycobacterium tuberculosis. Therefore, a diagnosis of miliary tuberculosis-associated HPS was made. The patient was successfully treated with antituberculous therapy.

A Community-acquired Lung Abscess Attributable to Streptococcus pneumoniae which Extended Directly into the Chest Wall.

We herein report the case of 75-year-old Japanese female with a community-acquired lung abscess attributable to Streptococcus pneumoniae (S. penumoniae) which extended into the chest wall. The patient was admitted to our hospital with a painful mass on the left anterior chest wall. A contrast-enhanced chest computed tomography scan showed a lung abscess in the left upper lobe which extended into the chest wall. Surgical debridement of the chest wall abscess and percutaneous transthoracic tube drainage of the lung abscess were performed. A culture of the drainage specimen yielded S. pneumoniae. The patient showed a remarkable improvement after the initiation of intravenous antibiotic therapy.