Thymic MALT lymphoma associated with Sjögren's syndrome with postoperative cardiac tamponade and acute pleuritis: a case report.

BACKGROUND: Thymic mucosa-associated lymphoid tissue (MALT) lymphoma is rare and is known to be associated with Sjögren's syndrome (SjS). SjS is rarely accompanied by serositis. Here, we describe the first case of postoperative cardiac tamponade and acute pleuritis in a patient with thymic MALT lymphoma associated with SjS. CASE PRESENTATION: A 33-year-old woman with SjS presented with an anterior mediastinal mass on chest computed tomography, which was performed for further examination of the condition. Suspecting a thymic MALT lymphoma or thymic epithelial tumor, total thymectomy was performed. The mediastinal mass was histopathologically diagnosed as a thymic MALT lymphoma. The patient was discharged with a good postoperative course but visited the hospital 30 days after surgery for dyspnea. Cardiac tamponade was observed and drainage was performed. Four days after pericardial drainage, chest radiography revealed massive left pleural effusion, and thoracic drainage was performed. The patient was diagnosed with serositis associated with SjS and treated with methylprednisolone, which relieved cardiac tamponade and pleuritis. CONCLUSIONS: Surgical invasion of thymic MALT lymphomas associated with SjS may cause serositis. Postoperative follow-up should be conducted, considering the possibility of cardiac tamponade or acute pleuritis due to serositis as postoperative complications.

Pleural Diseases in Connective Tissue Diseases.

Connective tissue diseases (CTD) are heterogeneous, immune-mediated inflammatory disorders often presenting with multiorgan involvement. With the advent of high-resolution computed tomography, CTD-related pleuritis-pleural thickening and effusion-is now increasingly recognized early in the disease trajectory. The natural history of CTD-related pleural effusions varies from spontaneous resolution to progressive fibrothorax with ventilatory impairment. Treatment of the underlying CTD is necessary to manage the pleural disease. Depending on the degree of symptom burden and physiological insult, specific treatment of pleural disease can include monitoring, repeated aspirations, systemic anti-inflammatory medication, and surgical decortication.

Is there a common definition for massive pleurisy?

The concept of massive pleurisy (MP) is frequently used to emphasize the significance of the amount of pleural effusion. However, there are significant disagreements about it due to the lack of a universal definition for MP. In our study, we sought to elucidate these distinctions. We employed a questionnaire comprised of visual and true/false sections. In the visual section, participants were shown real-time lung radiographs and schematic drawings and asked which ones were MP. On the other hand, suggestions regarding diagnosis, treatment, and consultations for MP were questionnaired. The study was comprised of 150 physicians from four distinct centers. On true/false and radiograph questions, physicians from the same branch exhibited differences of up to 50% (p < 0.05). On the level question, each branch involved reached a consensus (p = 0.003). In questions 3, 4, and 5, which also contained a true-false section, the branches gave varying responses, with the exception of the opinion that tube thoracostomy is unquestionably indicated in MP (p < 0.05). Establishing a common language for MP is crucial for clinician collaboration and appropriate patient management. Our study elucidates the divergences of opinion between branches and highlights the need for a unified definition.

Systemic lupus erythematosus in pediatric patients: Pulmonary manifestations.

The pulmonary manifestations of Systemic Lupus Erythematosus (SLE) in pediatric patients are poorly understood and the pulmonary manifestations reported from the adult population are generally extrapolated to the pediatric population. In the present work, the review of 228 files was carried out, in which the pulmonary manifestations, symptoms and antibody levels of the patients treated at the Hospital Regional de Alta Especialidad de Ixtapaluca (HRAEI), State of Mexico, Mexico, were identified. Statistical significance between groups was estimated using the Chi-square and Mann-Whitney U test. The main pulmonary manifestations identified were pleurisy (14 %), pulmonary hemorrhage (3.9 %), pulmonary thromboembolism (0.9 %), acute lupus pneumonitis (0.4 %), pulmonary arterial hypertension (0.4 %), and small lung syndrome (0.4 %). While the initial symptomatology was dyspnea with an incidence of 9.6 %, the mean oxygen saturation in the population was 96.87 %. Pleural effusion was identified as the most frequent pulmonary manifestation in radiographic changes. No statistically significant difference was found in antibody levels when comparing the groups. The most common pulmonary manifestation associated with SLE is pleurisy, however, the range of pulmonary manifestations in this type of patient can be very varied, as well as the presentation of each of them.

High prevalence of upper lung field pulmonary fibrosis radiologically consistent with pleuroparenchymal fibroelastosis in patients with round atelectasis.

BACKGROUND: Upper-lung field pulmonary fibrosis (upper-PF), radiologically consistent with pleuroparenchymal fibroelastosis (PPFE), was reported to develop in patients with a history of asbestos exposure and tuberculous pleurisy, indicating that chronic pleuritis is correlated with upper-PF development. Round atelectasis reportedly emerges after chronic pleuritis. This study aimed to clarify the association between round atelectasis and upper-PF. METHODS: We examined the radiological reports of all consecutive patients with round atelectasis between 2006 and 2018 and investigated the incidence of upper-PF development. RESULTS: Among 85 patients with round atelectasis, 21 patients (24.7%) were confirmed to finally develop upper-PF lesions. Upper-PF was diagnosed after round atelectasis recognition in more than half of the patients (13/21, 61.9%), whereas upper-PF and round atelectasis were simultaneously detected in the remaining 8 patients. At the time of round atelectasis detection, almost all patients (19/21, 90.5%) had diffuse pleural thickening and round atelectasis was commonly observed in non-upper lobes of 19 patients (90.5%). Fourteen patients had round atelectasis in unilateral lung, and the remaining 7 patients had round atelectasis in bilateral lungs. Among all 14 patients with unilateral round atelectasis, upper-PF developed on the same (n = 11) or both sides (n = 3). Thus, upper-PF emerged on the same side where round atelectasis was present (14/14, 100%). The autopsy of one patient revealed a thickened parietal-visceral pleura suggestive of chronic pleuritis. Subpleural fibroelastosis was also observed. CONCLUSIONS: Upper-PF occasionally develops on the same side of round atelectasis. Upper-PF may develop as a sequela of chronic pleuritis.

A case of synchronous IgG4-associated pleuritis and type 1 autoimmune pancreatitis.

A 50-year-old man presented to the emergency department with left chest pain, epigastralgia, and low-grade fever for several days. A CT scan showed left pleural effusion, ground-glass opacities in the lower lobes of both lungs, and a capsule-like rim in the pancreas. ERCP showed narrowing of the main pancreatic duct. EUS-FNA was performed, but pathological findings showed no IgG4-positive cells. A thoracoscopic biopsy was performed, and pathological findings showed many IgG4-positive cells. A diagnosis of autoimmune pancreatitis and IgG4-associated pleurisy was made according to international diagnostic criteria. After that, oral steroid therapy was started, and left pleural effusion and pancreatic enlargement improved.

[Chemotherapy-Resistant Breast Cancer and Carcinomatous Pleuritis Successfully Treated with Abemaciclib plus Letrozole Therapy].

A 78-year-old woman was examined in the outpatient department with a chief complaint of swelling of the left breast. Examination confirmed a 10 cm mass in the left breast as along with edema and redness of the skin, following which a diagnosis of invasive micropapillary carcinoma was made after biopsy. The CT imaging showed left chest wall invasion, multiple axillary lymph node metastases, and left carcinomatous pleuritis. Since this a case of advanced breast cancer, we initiated treatment with bevacizumab plus paclitaxel. After 8 months, her medication was changed to eribulin, owing to progression of the cancer, which continued even up to 4 months. We then initiated abemaciclib plus letrozole therapy as the third treatment. We observed tumor reduction and clearing of pleural effusion with no serious adverse events, and continued her therapy for 11 months before the cancer progressed. We report a case of chemotherapy-resistant breast cancer and carcinomatous pleuritis in an older adult patient for which abemaciclib plus letrozole therapy was effective.

[Pulmonary re-expansion in metastatic pleural effusions after thoracentesis: A pilot study]. / Échogénicité des pleurésies : facteur prédictif d'amélioration de la dyspnée ? Étude pilote concernant les pleurésies métastatiques.

INTRODUCTION: Metastatic pleural effusion is a cause of dyspnea. The American thoracic society has strongly suggested that studies evaluating thoracic ultrasonography as potentially predictive of improvment of dyspnea are needed. METHODS: We conducted a prospective monocentric observational study to assess chest ultrasound predictors of response to thoracentesis. Fifteen patients with metastatic pleural effusion were included. RESULTS: The initial mean VAS score was5 ± 2,9 cm. The majority of patients had pleural effusions equal to or greater than 5 intercostal spaces (EIC) in height, while 7 patients had an abnormal curvature of the hemidiaphragm (flattened or inverted). PRIMARY ENDPOINT: The volume removed was greater in the group with anechoic pleurisy compared to the group with sonographic septation, notwithstanding complex pleural effusion (non-septated, relatively hyperechoic, with some spots in the effusion). The patients with complex pleural effusions had an higher score of dyspnea. SECONDARY ENDPOINTS: The 7 patients with abnormal diaphragmatic curvature presented significant dyspnea with a pain score of approximately 7 and profuse pleurisy occupying 8 intercostal spaces in height. The effusions of those who could not normalize their curvature had a complex aspect and the volume removed was lower. CONCLUSIONS: The ultrasound characteristics of pleural effusions seem to be predictors of improvment of dyspnea after thoracentesis. The septated and complex aspects are probably predictors of non improvment of dyspnea.

Transient and Recurrent Pulmonary Infiltrations Associated with Familial Mediterranean Fever.

Chest symptoms and pleural effusion due to serositis in familial Mediterranean fever (FMF) are occasionally misdiagnosed as acute pneumonia. However, the actual pulmonary involvement of FMF is extremely rare. A 67-year-old man was referred to our hospital due to repeated and transient anterior chest pain. Chest images revealed a moderate amount of pericardial fluid, slight bilateral pleural effusion, and infiltrations in both lower lung lobes. Colchicine treatment without antibiotics rapidly improved these symptoms and findings. Pericarditis, pleurisy and the response to colchicine indicated FMF. FMF should be considered as a causative disease of pulmonary infiltrations, especially if it occurs repeatedly.

[A Case of Late Recurrence of Breast Cancer Causing Carcinomatous Pleurisy 29 Years Postoperatively].

The patient was a 81-year-old female with a history of treatment for the right breast cancer 29 years earlier. She presented with a chief complaint of dyspnea. CT showed pleural effusion and a mediastinal tumor. A biopsy was performed on the mediastinal tumor, and not only pathological but also immunohistological examination findings were similar to those of the surgical specimens 29 years ago. In view of the patient's age, we initiated treatment by anastrozole. The treatment was effective, and other forms of endocrine therapy were administered. She continued to be treated with the endocrine therapy over 4.5 years. The late recurrence of breast cancer in the form of carcinomatous pleurisy 29 years postoperatively is rare.

An unusual case of explosive pleuritis without mediastinal shift.

Explosive pleuritis (EP) is an under-reported condition. Although there is no agreed-upon definition, generally, the term EP denotes a dramatic clinical and radiologic progression of pleural space inflammation in the setting of pneumonia. The pleural space inflammation leads to the formation of loculated complex pleural effusion or empyema, and pleural adhesion within a matter of hours, typically in less than 24 hours. Radiologically, these changes are manifested as opacification of the hemithorax with or without contralateral mediastinal shift. Streptococcal pneumonia is the most common cause of EP. Here we present a case and discuss the clinical, microbiologic, radiologic, and treatment aspects of EP. We also scrutinize the medical literature to compare the previously reported cases.

Marked increase of interferon-ß after BNT162b2 mRNA vaccination: a case of polyarthritis with pleurisy.

Exacerbation of rheumatic disease after vaccination against SARS-CoV-2 is being reported. However, there are only a few cases of new-onset rheumatic diseases. We present two cases of new-onset persistent polyarthritis that developed in patients after receiving the mRNA vaccine against SARS-CoV-2. One patient had bilateral pleural effusions with markedly elevated serum interferon (IFN)-ß, while the other had no effusion, with serum IFN-ß comparable with that in healthy subjects. Other cytokines were unaltered in association with effusion. Both patients responded well to treatment with 20 mg prednisolone. Although more investigations are needed, the marked increase in serum IFN-ß levels observed in the case with pleural effusion may reflect an excessive response from the innate immune system to mRNA vaccines.

Epidemiologic evaluation of pleurisy diagnosed by surgical pleural biopsy using data from a nationwide administrative database.

BACKGROUND: Pleural biopsies for investigating the causes of pleurisy are performed through modalities including needle biopsies, local anesthetic thoracoscopic procedures, and surgery (video-assisted thoracoscopic surgery and open thoracotomy). To date, there have been no large-scale nationwide epidemiological studies regarding pleurisy diagnosed via surgical pleural biopsy. This study examined the epidemiology of pleurisy diagnosed via surgical pleural biopsy in a Japanese nationwide administrative database. METHODS: We evaluated Japanese Diagnosis Procedure Combination data of 24 173 patients who underwent video-assisted thoracoscopic surgery or open thoracotomy and received a diagnosis of pleurisy between April 2014 and March 2020. In addition to pleurisy diagnoses, the patients' clinical information, including age, sex, smoking status (pack-years), dyspnea grade, length of in-hospital stay, and comorbidities, were extracted from the dataset. RESULTS: This study included data from 1699 patients. The most frequent causes of pleurisy were neoplastic diseases (55.9%; malignant mesothelioma 22.5%, lung cancer 15.7%, lymphoma 2.5%), followed by infectious diseases (24.0%; tuberculosis 16.2%, parapneumonic pleural effusion 3.6%, empyema 3.5%, nontuberculous mycobacteriosis 0.5%), collagen vascular diseases (2.8%; rheumatoid arthritis 1.3%, immunoglobulin G4-related diseases 0.7%, systemic lupus erythematosus 0.3%), and paragonimiasis (0.1%). CONCLUSIONS: Neoplastic diseases, including malignant mesothelioma and lung cancer, were frequently and accurately diagnosed as pleurisy via surgical pleural biopsy. The next leading cause was infectious diseases such as mycobacterial infections. Physicians should consider performing surgical biopsy in light of the knowledge regarding the etiology of pleurisy when a definitive diagnosis cannot be made via needle pleural biopsy.

Concomitant pleuritis and pericarditis developing during glucocorticoid therapy: a case report on granulomatosis with polyangiitis.

Granulomatosis with polyangiitis is an anti-neutrophil cytoplasmic antibody-associated vasculitis that manifests in various ways by affecting the small-sized vessels in multiple organs. Acute pleuritis and pericarditis are both rare among the different manifestations of granulomatosis with polyangiitis. The symptoms in each of the organs are often apparent at the time of diagnosis and tend to diminish with treatment. Organ damage and progression of the disease during treatment are uncommon. We encountered a patient with granulomatosis with polyangiitis who, after starting intravenous methylprednisolone pulse therapy, concurrently developed acute pleuritis and pericarditis. The patient was a 47-year-old Japanese man with myalgia in whom kidney dysfunction, proteinase 3-anti-neutrophil cytoplasmic antibody positivity, and a lung mass were detected. Granulomatosis with polyangiitis was diagnosed pathologically from a lung and a kidney biopsy. Acute pleuritis and pericarditis, which developed after the first course of intravenous methylprednisolone pulse therapy, both resolved following the second course. The present report indicates that secondary serositis such as pleuritis and pericarditis can develop in patients with granulomatosis with polyangiitis even during glucocorticoid therapy.

[Anterior Mediastinal Cholesterin Granuloma Associated with Bilateral Pleurisy and Pericarditis].

The patient was a 41-year-old man. He was diagnosed with pleurisy and came to our hospital. The pleural effusion and pleurisy remained even after administration of sufficient doses of antibiotics. A thorough examination revealed an anterior mediastinal tumor. Six months later, pericarditis also developed. Autoimmune diseases, infections, and malignant diseases were suspected, but a definitive diagnosis could not be made. In order to confirm the diagnosis, anterior mediastinal tumor resection and pleural biopsy were performed. The anterior mediastinal tumor was diagnosed as cholesterin granuloma pathollogically. Cholesterin granuloma is a granuloma formed by deposition of cholesterin crystals and cholesterin granuloma occurring in the mediastinum is extremely rare.

ANA positivity and complement level in pleural fluid are potential diagnostic markers in discriminating lupus pleuritis from pleural effusion of other aetiologies.

OBJECTIVE: Lupus pleuritis is the most common pulmonary manifestation of systemic lupus erythematosus (SLE). We aimed to compare various biomarkers in discriminating between pleural effusions due to lupus pleuritis and other aetiologies. METHODS: We determined in 59 patients (16 patients with SLE and 43 patients without SLE) pleural fluid levels of high-mobility group box 1, soluble receptor for advanced glycation end products (sRAGE), adenosine deaminase (ADA), interleukin (IL) 17A, tumour necrosis factor-α, antinuclear antibodies (ANA), and complements C3 and C4. RESULTS: We found significant differences in the pleural fluid level of sRAGE, ADA, IL-17A, C3 and C4, and in the proportion of ANA positivity, among lupus pleuritis and other groups with pleural effusion. Specifically, ANA positivity (titre ≥1: 80) achieved a high sensitivity of 91%, specificity of 83% and negative predictive value (NPV) of 97% in discriminating lupus pleuritis from non-lupus pleural effusion. A parallel combination of the level of C3 (<24 mg/dL) and C4 (<3 mg/dL) achieved a sensitivity of 82%, specificity of 89% and NPV of 93% in discriminating lupus pleuritis from non-lupus exudative pleural effusion. CONCLUSIONS: In conclusion, ANA, C3 and C4 in pleural fluid are useful in discriminating lupus pleuritis from pleural effusion due to other aetiologies with high NPV.

An Elderly Male with Primary Sjögren's Syndrome Presenting Pleuritis as the Initial Manifestation.

Primary Sjögren's syndrome (SS) is an autoimmune disease that usually affects the exocrine glands in mid-dle-aged women. Fifteen percent of SS patients experience severe systemic extraglandular complications, and pleuritis is one of the rare complications of SS. We report the case of an elderly Japanese man who initially pre-sented with a prolonged fever and chest pain and was finally diagnosed with primary SS-associated pleuritis. Of the nine reported cases of primary SS that initially presented with pleuritis, up to six cases were elderly males. This case highlights the complication of pleuritis among elderly males with primary SS.