Chylothorax in Henoch-Schonlein purpura: a case report and review of the literature

Henoch-Schonlein purpura (HSP) is the most common acute vasculitis in the pediatric population, with an incidence of 10-14 per 100,000. The classic presentation of this disorder includes erythematous papules followed by palpable purpura in the lower extremities, trunk, and face, arthralgia or arthritis, abdominal pain, gastrointestinal bleeding, and nephritis. While renal abnormalities in HSP are common, the classic pulmonary manifestations, such as hemorrhage and pneumonitis, are thought to be infrequent. Subclinical pulmonary manifestations, including diffusion defects and radiographic anomalies, seem to be quite frequent in patients with HSP but are not commonly reported. Other respiratory manifestations include pleural effusion and chylothorax, but these are rarely mentioned in the literature. Chylothorax was only reported once in an adult patient with HSP in whom the mechanism of formation was demonstrated to be secondary to transdiaphragmatic passage of chylous fluid from the peritoneal cavity. Here we describe an 8-year-old girl with HSP, nephrotic syndrome, and chylothorax, and we report the results of a review of the literature regarding respiratory complications in HSP. The present case is the first pediatric patient reported with HSP and chylothorax. The therapeutic measures utilized were effective in resolving her edema, ascites, and chylothorax, and we advocate the use of these measures as first-line therapy in future patients with similar complications from HSP.     

CD56-positive Angioimmunoblastic T-cell Lymphoma Complicated by Chylothorax

A 77-year-old woman presented with systemic lymphadenopathy and bilateral pleural effusion. Angioimmunoblastic T-cell lymphoma (AITL) was diagnosed based on the results of a lymph node biopsy. AITL cells expressed the aberrant antigen of CD56. The bilateral pleural effusion was attributed to chylothorax, not the infiltration of lymphoma cells into the pleura, as determined by the pleural fluid analysis. We therefore diagnosed her with CD56-positive AITL complicated by chylothorax. She achieved complete remission by multidrug chemotherapy. AITL is commonly complicated by pleural effusion, but rarely by chylothorax. This is the first case of CD56-positive AITL complicated by chylothorax.     

恶性胸膜间皮瘤致嗜酸细胞性胸腔积液1例报告并文献复习

目的提高对嗜酸细胞性胸腔积液的认识。方法对一例由恶性胸膜间皮瘤所致嗜酸细胞性胸腔积液的临床资料进行分析,并结合文献复习。结果嗜酸细胞性胸腔积液首次报道于1984年,其病因很复杂,由肿瘤引起的并不多见,机理也不十分清楚,大多预后良好。结论虽然嗜酸细胞性胸腔积液的病因大多数是良性的,但我们不能忽略恶性的情况,应尽可能查找原因,以免贻误病情。     

多发性骨髓瘤伴胸膜浸润1例

1病历资料患者女,77岁。2010年2月起反复出现胸闷、气急,无发热、咳嗽、盗汗、胸痛、心悸等不适,夜间尚可平卧。既往有高血压病史20余年,血压最高达180/110 mmHg(1 mmHg=0.133 kPa),平素服用降压药,血压控制良好。入院体检:神志清楚,消瘦,全身皮肤无黄染,无皮下出血点,球结膜无水肿,颈     

Primary pleural epithelioid hemangioendothelioma (EHE)--two cases and review of the literature

Introduction: We present two cases with symptoms of progressively worsening cough, dyspnea, decreased exercise tolerance and right‐sided back pain in the first case and upper respiratory symptoms characterized by cough and a low grade fever in the second case. Methods: Report of two cases. Results: The initial chest X‐ray in both the cases showed pleural effusion. Further imaging with computed tomography of the chest confirmed the effusion in both cases. Thoracentesis was done in both of them revealed an exudative effusion that did not reveal any infection or malignancy. Both cases underwent surgical biopsy and the diagnosis of primary pleural epithelioid hemangioendothelioma was made. Conclusions: Both the cases had progressive clinical deterioration despite chemotherapy with Taxol and Bevacizumab in one case and carboplatin, etoposide, and bevacizumab, in the second case. Both developed metastatic disease to lungs and died. Please cite this paper as: Lazarus A, Fuhrer G, Malekiani C, McKay S and Thurber J. Primary pleural epithelioid hemangioendothelioma (EHE) – two cases and review of the literature. Clin Respir J 2011; 5: e1–e5.     

Primary effusion lymphoma of the pleural space: Report of a rare complication of cardiac transplant with review of the literature

Primary effusion lymphoma (PEL) is a rare mature B‐cell non‐Hodgkin’s lymphoma arising in body cavities and presenting with effusions. It has been described predominantly in patients with impaired immunity from the acquired immunodeficiency syndrome and is associated with the Human Herpesvirus‐8 (HHV‐8). Seldom has PEL been diagnosed in persons negative for the human immunodeficiency virus (HIV), and in such cases it has occurred primarily in the setting of posttransplant immunosuppression. We report an instructive case of a Caribbean‐American HIV‐negative orthotopic heart transplant recipient with a history of HHV‐8‐associated Kaposi's sarcoma who developed HHV‐8 viremia and PEL of the pleural space early in the posttransplant course. This case highlights the importance of considering PEL in the differential diagnosis of a new pleural effusion in a transplant recipient at risk for HHV‐8‐associated disease.     

Pleural tuberculosis: A concise clinical review

Tuberculosis (TB) is the leading infectious cause of death worldwide, and the commonest cause of death in people living with HIV. Globally, pleural TB remains one of the most frequent causes of pleural exudates, particularly in TB‐endemic areas and in the HIV positive population. Most TB pleural effusions are exudates with high adenosine deaminase (ADA), lymphocyte‐rich, straw‐coloured and free flowing, with a low yield on mycobacterial culture. TB pleurisy can also present as loculated neutrophil‐predominant effusions which mimic parapneumonic effusions. Rarely, they can present as frank TB empyema, containing an abundance of mycobacteria. Up to 80% of patients have parenchymal involvement on chest imaging. The diagnosis is simple if M. tuberculosis is detected in sputum, pleural fluid or biopsy specimens, and the recent advent of liquid medium culture techniques has increased the microbiological yield dramatically. Where the prevalence of TB is high the presence of a lymphocyte‐predominant exudate with a high ADA has a positive predictive value of 98%. In low prevalence areas, the absence of an elevated ADA and lymphocyte predominance makes TB very unlikely, and pleural biopsy should be performed to confirm the diagnosis. Pleural biopsy for liquid culture and susceptibility testing must also be considered where the prevalence of drug resistant TB is high. Treatment regimens are identical to those administered for pulmonary TB. Initial pleural drainage may have a role in symptom relief and in hastening the resolution of the effusion. Surgical intervention may be required in loculated effusions and empyemas.     

A case report of IgG4-related respiratory disease with pleural effusion and a literature review

Rationale:IgG4-related respiratory disease (IgG4-RRD) is a chronic autoimmune disease that affects the respiratory system and organs outside the respiratory system. This study explored the diagnosis and treatment of a case of IgG4-RRD with unilateral pleural effusion diagnosed using medical thoracoscopy, and provides an associated literature review. This report summarizes the clinical characteristics of IgG4-RRD involving the pleura to improve the diagnosis of this disease.Patient concerns:A 39-year-old man presented with a 2-week history of cough and chest tightness. Both physical examination and imaging supported the presence of left pleural effusion.Diagnosis:Medical electronic thoracoscopy was performed to obtain a pleural biopsy, which showed lymphoplasmacytic infiltration, 40 IgG4+ plasma cells per High Power Field (HPF) on microscopy, IgG4/IgG ratio >50%, phlebitis obliterans, and storiform fibrosis. The final diagnosis was IgG4-RRD.Interventions and outcomes:The patient was treated with methylprednisolone, after which his symptoms improved, and he was discharged. Oral hormone therapy was continued outside the hospital. After 4 months, the patient returned to the hospital and his condition had improved significantly.Lessons:Pleural involvement in IgG4-RRD is rare, and its diagnosis depends on pleural biopsy. Thoracoscopy usually reveals pleural thickening, pleural nodules, and milky white plaques.     

Description of a new technique: management of pleural effusion with a Cystofix catheter

Pleural effusion (PE) is a common and serious complication of respiratory disease that often requires drainage. Forty-four patients whose PE were successfully treated with a Cystofix catheter are reported. This is a new technique for draining PE that should be considered in all patients.     

Prevalence and characteristics of pleural effusions in superior vena cava syndrome

OBJECTIVE AND BACKGROUND: The prevalence and characteristics of pleural effusions occurring in adults with the superior vena cava (SVC) syndrome are unknown. The purpose of the present study was to characterize these pleural effusions. METHODS: Charts of patients diagnosed with SVC syndrome at a tertiary care referral centre were reviewed. Radiographs were evaluated for the presence and size of pleural effusions, presence and location of masses and mediastinal width. If a pleural effusion was present, the patient's chart and a pre-existing database on pleural effusions were searched to determine whether the effusion was sampled and the results of any laboratory investigations on the fluid. RESULTS: The SVC syndrome occurred in 78 patients. Malignancy was the aetiology in 60% of the cases and bronchogenic carcinoma was the most common malignancy. An intravascular device was the aetiology in the majority of benign cases. Pleural effusion was found in 70% of patients with a malignant aetiology and 58% of those with a benign cause (P=0.345). The mean size of the effusions was larger in malignant cases (P=0.012). Of the 44 effusions 22 were sampled (17 in malignancy and five with benign processes); none was transudates, 20 (91%) were exudative (four of these were chylous) and the remaining two were reported as exudates but did not have pleural chemistries documented. CONCLUSIONS: More than half of patients with SVC syndrome have pleural effusions, regardless of the aetiology. However, the effusions are larger when associated with malignancy. The majority of these effusions are exudative and occasionally chylous. None was transudates.     

Diagnosis and management of malignant pleural effusions

Abstract:   Malignant pleural effusions (MPEs) complicate the clinical course of patients with a broad array of malignancies, which are most often due to lymphomas or carcinomas of the breast, lung, gastrointestinal tract or ovaries. Patients may present with a MPE as the initial manifestation of a cancer or develop an effusion during the advanced phases of a known malignancy. In either circumstance, the median survival after presentation with a MPE is 4 months. Effusions may result from direct pleural invasion (MPE) or indirect effects (paraneoplastic effusions), such as impairment of fluid efflux from the pleural space by lymphatic obstruction or pleural effects of cancer radiation or drug therapy. Because only 50% of patients with cancer who develop a pleural effusion during their clinical course have a MPE, careful evaluation of the effusion to establish its aetiology is required to direct therapy. Management is palliative with interventions directed towards decreasing the volume of intrapleural fluid and the severity of associated symptoms.     

Pleural procedural complications: prevention and management

Pleural disease is common with a rising case frequency. Many of these patients will be symptomatic and require diagnostic and/or therapeutic procedures. Patients with pleural disease present to a number of different medical specialties, and an equally broad range of clinicians are therefore required to have practical knowledge of these procedures. There is often underestimation of the morbidity and mortality associated with pleural interventions, even those regarded as being relatively straightforward, with potentially significant implications for processes relating to patient safety and informed consent. The advent of thoracic ultrasound (TUS) has had a major influence on patient safety and the number of physicians with the necessary skill set to perform pleural procedures. As the variety and complexity of pleural interventions increases, there is increasing recognition that early specialist input can reduce the risk of complications and number of procedures a patient requires. This review looks at the means by which complications of pleural procedures arise, along with how they can be managed or ideally prevented.     

Diagnostic accuracy of interleukin-33 for tuberculous pleural effusion: A systematic review and meta-analysis

Background:The detection of interleukin 33 (IL-33) in pleural effusion may be more sensitive in diagnosing tuberculous pleural effusion (TPE). The present study aimed to assess the accuracy of pleural IL-33 for the diagnosis of TPE by means of meta-analysis and systematic review of relevant studies.Method:After retrieving the published studies, the sensitivity, specificity, positive likelihood ratio, negative likelihood ratio, diagnostic odds ratio, and a summary receiver operating characteristic curve were assessed to estimate the usefulness of pleural IL-33 in diagnosing TPE using meta-analysis with a random-effects model. We also performed meta-regression and subgroup analysis.Results:A total of 639 patients from 6 studies were analyzed. The pooled sensitivity, specificity, positive likelihood ratio, negative likelihood ratio, and diagnostic odds ratio were 0.87 (95% confidence interval [CI], 0.82–0.91), 0.76 (95% CI, 0.72–0.80), 6.54 (95% CI, 2.65–16.15), 0.17 (95% CI, 0.10–1.27), and 45.40 (95% CI, 12.83–160.70) respectively. The area under the curve was 0.94. The composition of the included population was the main cause of heterogeneity and subgroup analysis showed that pleural IL-33 had a higher specificity (0.93, 95% CI 0.87–0.96) when used for differential diagnosis between TPE and malignant pleural effusion.Conclusion:The detection of IL-33 alone in pleural effusion seems to not be an efficient diagnostic marker for TPE but may serve as a novel biomarker to differentiate between TPE and malignant pleural effusion.     

Non‐Hodgkin lymphoma manifesting as massive malignant chylothorax: successful management with chemotherapy and ambulatory drainages using indwelling pleural catheter

Recurrent cancer‐related chylothorax is generally managed by talc pleurodesis or indwelling pleural catheter in the palliative care setting to relieve symptoms and improve quality of life. In chylothorax associated with curable/treatable malignancies like lymphoma, there are scarce data regarding the efficacy and safety of indwelling pleural catheters. We report a case of recurrent massive chylothorax associated with non‐Hodgkin lymphoma who demonstrated long‐term remission of lymphoma and complete regression of chylothorax after treatment with combination chemotherapy and ambulatory drainages using indwelling pleural catheter.