[Case of pulmonary inflammatory pseudotumor with cysts].

We herein report a case involving a 58-year-old female patient with multiple cystic lesions in the right lobe of the lung. The lesions were revealed on chest computed tomography in 2002 and followed up. Transbronchial lung biopsy showed no malignancy in June 2013. The lesions gradually increased in size and thickness and were associated with fluid-filled cysts. We performed a right lower lobectomy in November 2013. Pathological examination revealed inflammatory pseudotumor. Such a case of inflammatory pseudotumor presenting as a pulmonary cyst has not been previously described. Intractable infection and inflammation are regarded as common causes of inflammatory pseudotumor. This condition should be considered in patients with a medical history consistent with infectious disease and a pulmonary cyst found on chest computed tomography.

Pulmonary inflammatory myofibroblastic tumour after Hodgkin's lymphoma.

Positron emission tomography (PET)/CT plays a major role in staging, assessing response to treatment and during follow-up of paediatric Hodgkin's lymphoma (HL). Owing to high sensitivity to detect viable tumoural tissue, negative PET/CT is highly predictive of survival. However, (18)F-FDG is not specific for malignant disease and may concentrate in numerous benign/inflammatory lesions that may cause 'false-positive' results and follow-up PET/CT studies should be interpreted with caution. We report a case of pulmonary inflammatory myofibroblastic tumour, which developed during follow-up in a young patient with complete remission of a stage IIB HL and was fully treated with surgical resection.

[Pseudotumor phenotype of sarcoidosis: about two cases]. / Phénotype pseudotumoral de la sarcoïdose: à propos de deux cas.

INTRODUCTION: Sarcoidosis is a multisystem benign granulomatous disease of unknown etiology. It can sometimes cause diagnostic confusion by presenting in the form of a pseudotumor, thus constituting a trap for the unwary. COMMENTS: The authors report two cases of pseudotumor sarcoidosis. In the first, the pseudotumor occurred in the context of multisystem disease in a 48-year-old man. The response to treatment with systemic corticosteroids was complicated by the development of disseminated tuberculosis, which was rapidly fatal. The second case, by contrast, was about a 58-year-old woman and the disease was self-limiting, resolving spontaneously in less than 3 months. CONCLUSION: Through these two cases, the authors focus on the particularities of the pseudotumor manifestation of pulmonary sarcoidosis. The diagnosis is often difficult. The mismatch between the clinical picture and the extent of radiological lesions should, however, suggest the diagnosis. Histological evidence is needed to eliminate other etiologies including malignant tumors. The condition usually resolves either spontaneously or after treatment with systemic corticosteroids. However, relapses are possible, including on discontinuation of corticosteroid therapy.

Postradiofrequency ablation inflammatory pseudotumor associated with pulmonary venoocclusive disease: case report and review of the literature.

Radiofrequency ablation of pulmonary veins is a common therapeutic intervention for atrial fibrillation. Pulmonary vein stenosis and venoocclusive disease are recognized complications, but the spectrum of pathologies postablation have not been previously reviewed. A recent case at our hospital showed a left hilar soft tissue mass in association with superior pulmonary vein stenosis in a patient 4 years postablation. On resection, this proved to be an inflammatory pseudotumor composed of myofibroblasts in an organizing pneumonia-type pattern with adjacent dendriform ossifications. Pulmonary venoocclusive change was a prominent feature. Literature on the histopathology of postradiofrequency ablation complications is limited. The severity of vascular pathology appears to increase with the postablation interval. Although pulmonary vascular changes are the most common late finding, fibroinflammatory changes including pulmonary pseudotumor formation, attributable to thermal injury, should be considered in the differential diagnosis of these cases.

Resolución espontánea de un seudotumor inflamatorio del pulmón como consecuencia de una biopsia en cuña / Spontaneous Resolution of an Inflammatory Pseudotumour of the Lung Subsequent to Wedge Biopsy

La resolución completa y espontánea de un seudotumor inflamatorio (STI) de los pulmones es excepcional. Un hombre de 44 años de edad fue derivado para la valoración de una «neumonía no resuelta». Refería tos y expectoración mínima durante 5 meses, al igual que dolor torácico, hemoptisis y fiebre durante 2 semanas. La tomografía computarizada (TC) de tórax confirmó la presencia de una masa homogénea de bordes irregulares localizada en el lóbulo medio derecho con áreas de dispersión y broncograma aéreo. La biopsia pulmonar transbronquial era sugestiva de un «granuloma de células plasmáticas». En la toracotomía se puso de relieve una masa dura localizada en el lóbulo medio derecho y adherida al lóbulo inferior, la pared torácica y el mediastino, cuya resección no fue posible. Una biopsia en cuña confirmó un STI. La radiografía de tórax, efectuada 4 semanas después, reveló una resolución sustancial. En la TC de tórax, realizada un año más tarde, se demostró una escara fibrótica. Hasta la fecha solo se han publicado 6 casos de pacientes con una resolución espontánea de PTI de los pulmones, y en 4 se describió al cabo de 3 meses de una intervención diagnóstica cruenta(AU)

Spontaneous, complete resolution of inflammatory pseudotumour (IPT) of lungs is exceptionally rare. A 44-year-old male was referred for evaluation for «non resolving pneumonitis». He had cough and minimal expectoration for 5 months, chest pain, haemoptysis and fever for a fortnight. Computed tomography of thorax (CT-thorax) confirmed the presence of a homogenous mass with irregular borders in right middle lobe with areas of breakdown and air bronchogram. Transbronchial lung biopsy was suggestive of «plasma cell granuloma». Thoracotomy disclosed a hard mass in right middle lobe adherent to lower lobe, chest wall and mediastinum which could not be removed. A wedge biopsy confirmed IPT. Chest radiograph after 4 weeks revealed significant resolution. CT-thorax a year later showed fibrotic scar. Till date, there are only five reports documenting 6 patients with spontaneous resolution of IPT of lungs and in 4 this occurred within 3 months of an invasive diagnostic intervention(AU)

Granuloma pulmonar hialinizante / Pulmonary Hyalinizing Granuloma

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[Traumatic pulmonary pseudocyst: 2 case reports]. / Travmatik pulmoner psödokist: Iki olgu sunumu.

Traumatic pulmonary pseudocysts (TPP) are cavitary lesions that are rarely seen after blunt thoracic traumas. Two male patients who were diagnosed with cystic lesions in the left lung after trauma were followed in our clinic with the diagnosis of TPP. Due to increase in cyst dimensions and wall tension, which were seen on the follow-up thorax tomography, surgical intervention was decided for both cases. The first case underwent cystotomy and capitonnage via thoracotomy, and was discharged without any complication. However, the second case was lost due to cardiac arrest during the operative preparations. Thorax tomography is an important method in the diagnosis and radiological follow-up of TPP. The surgery option should always be remembered for patients who show progression during the follow-up.

Pulmonary histoplasmosis producing a spindle cell "pseudotumor".

Pulmonary spindle cell proliferations have been reported in association with a limited group of infectious agents. These lesions are rare and identified most often in the setting of immunosuppression. Because their appearance can simulate a spindle cell neoplasm, they are diagnostically treacherous, sometimes delaying antimicrobial therapy or resulting in unnecessary surgery. We report a case of a spindle pseudotumor of the lung resulting from Histoplasma capsulatum infection, a previously unreported cause of a spindle cell lesion in the lung. The patient was a 67-year-old woman in whom positron emission tomography-positive nodules developed in the left lung and left mediastinum. The patient had undergone renal transplantation and was receiving immunosuppressive therapy with mycophenolate, tacrolimus, and low-dose prednisone. Infection with H capsulatum was confirmed by culture of pleural effusion fluid, DNA probe analysis of the pleural fluid culture isolate, urinary Histoplasma antigen detection, and Grocott methenamine silver stains of tissue sections. To our knowledge, this is the first case of a spindle cell "pseudotumor" of the lung resulting from histoplasmosis. It highlights the importance of performing special stains for organisms when evaluating pulmonary spindle cell lesions in an immunocompromised host.

Complication of bovine pericardial buttress: pulmonary pseudotumor.

A 38-year-old man reported progressive back pain 4 years after undergoing partial resection of the lung for spontaneous pneumothorax, using staples buttressed with bovine pericardium. Chest computed tomography detected a mass near the staple line. Resection of the mass was performed successfully and the pain was relieved. The excised material was identified as an inflammatory pulmonary pseudotumor caused by the buttressing material.

Treatment with bortezomib of a patient having hyper IgG4 disease.

Hyper IgG4 disease is a recently described inflammatory disease characterized by lymphoplasmacytic infiltration leading to fibrosis and tissue destruction. Whereas most cases have been successfully treated with corticosteroids, recurrent or refractory cases may benefit from alternative therapies. Bortezomib has proven to be successful in the treatment of multiple myeloma, and its mechanism indicates that it may have merit in autoimmune or other plasmacytic disorders. We report a patient with recurrent pulmonary infiltration with IgG4 plasma cells, consistent with hyper IgG4 disease, who was successfully treated using a bortezomib-based combination with minimal therapy-related toxicities.

Pseudocyst in pulmonary acute respiratory distress syndrome (ARDS).

Childhood ARDS is mostly caused by pneumonia. Pulmonary pseudocysts are reported in adults recovering from ARDS, usually in non-dependent lung regions. We present a 1.5-year-old boy, who survived severe pulmonary ARDS with development of pulmonary giant pseudocysts and other structural abnormalities in dependent lung region. To the best of our knowledge, it is the first follow up report of pulmonary abnormality In a toddler with ARDS of extreme severity.

Variable expression of tenascin-C, osteopontin and fibronectin in inflammatory myofibroblastic tumour of the lung.

The aim of this study was to analyse the expression of tenascin-C, osteopontin and fibronectin in inflammatory myofibroblastic tumour of the lung, which is a rare tumour of unknown aetiology. Nine patients with an inflammatory myofibroblastic tumour of lung were studied by immunohistochemistry for the presence of tenascin-C, osteopontin, fibronectin and alpha-smooth muscle actin, which is a common marker for myofibroblasts. The ultrastructure of myofibroblasts was confirmed by transmission electron microscopy. The expression of tenascin-C, osteopontin, fibronectin and alpha-smooth muscle actin was also studied by immunoelectron microscopy. All cases displayed all of the studied extracellular matrix proteins and also alpha-smooth muscle actin-positive spindle-shaped fibroblastic cells that were undoubtedly myofibroblasts. The immunoelectron microscopic studies demonstrated labelling for alpha-smooth muscle actin in intracellular filament bundles within myofibroblasts, for fibronectin in the extracellular filaments of the fibronexus and for tenascin-C extracellularly often adjacent to myofibroblasts. Labels for osteopontin were observed within myofibroblasts and plasma cells. These results demonstrate that tenascin-C, osteopontin and fibronectin were expressed in all three kinds of subtypes of inflammatory myofibroblastic tumours of the lung and further, variable amounts of myofibroblasts could be observed by light and transmission electron microscopy as well as by immunoelectron microscopic techniques.

Traumatic pulmonary pseudocyst: a case report.

Traumatic pulmonary pseudocyst is a rare clinical event that may occur following chest trauma. This complication usually occurs as a result of blunt trauma and rarely, due to a penetrating injury. We report an unusual case of a 10-year-old boy who developed a left-sided pneumothorax along with a traumatic pulmonary pseudocyst when he was hit by a cricket ball while playing.

Pseudotumor associated with polytetrafluoroethylene sleeves.

We report the case of a patient who was operated on in February 2001. We performed a wedge resection of the upper right lobe. The pathologic examination demonstrated a lung adenocarcinoma (pT2N0M0, R0). We used staple line reinforcement material (ePTFE) during the operation because the patient had an important emphysema. We re-operated in January 2005 because during follow-up we observed a suspicious image that suggested a tumoral relapse. Histopathological study showed extrinsic material compatible with the one used in the original resection.

Pulmonary non-caseating granuloma in Waldenström macroglobulinemia.

Lung involvement is rare in Waldenström macroglobulinemia (WM). We encountered a male patient with WM who complained of breathlessness. Chest X-ray revealed diffuse infiltrative shadow throughout the both lungs. Transbronchial biopsy showed infiltration of atypical plasmacytoid lymphocytes and non-caseating granuloma. We treated the patients with fludarabine phosphate, and both his symptom and X-ray findings were then improved. To our knowledge, this is the first case showing non-caseating granuloma with lung involvement of WM. We discuss a mechanism of non-caseating granuloma formation in this case.

Endobronchial pseudo-tumour caused by herpes simplex.

Herpes simplex virus (HSV) causes tracheobronchitis and pneumonitis; however, to date, there has only been one report of an endobronchial mass caused by HSV type II. This case study describes a 68-yr-old female with severe kyphoscoliosis who was intubated for acute on chronic hypercapnic respiratory failure and developed blood-tinged endotracheal secretions. Fibreoptic bronchoscopy demonstrated an endobronchial mass in the right middle lobe. Cultures grew HSV type I and biopsy specimens demonstrated cytopathological changes consistent with HSV infection. This is the first reported case of HSV type I presenting as an endobronchial tumour.

Opposing roles for IL-13 and IL-13 receptor alpha 2 in health and disease.

Interleukin (IL)-13 is a key inducer of several type-2 cytokine-dependent pathologies. It regulates inflammation, mucus production, tissue remodeling, and fibrosis. Consequently, it has become an important therapeutic target for a number of debilitating illnesses, including asthma, idiopathic pulmonary fibrosis, ulcerative colitis, as well as several other diseases in which IL-13 is believed to be overproduced. In the murine model of schistosomiasis, IL-13 has emerged as a central mediator of chronic infection-induced liver pathology. Although IL-4, IL-5, IL-10, and IL-13 each regulate distinct aspects of the granulomatous inflammatory response, IL-13 was identified as the primary mediator of liver fibrosis. Thus, elucidating the mechanisms that regulate the production and function of IL-13 has become an intensive area of research. IL-13 signaling is mediated by the type-2 IL-4 receptor, which consists of the IL-4R alpha and IL-13R alpha 1 chains. However, another IL-13-binding chain, IL-13R alpha 2, appears to strongly inhibit the activity of IL-13. Animals deficient in IL-13R alpha 2 fail to downmodulate granuloma formation in the chronic phase of infection. They also develop severe IL-13-dependent fibrosis and portal hypertension and quickly succumb to the infection. Here, we summarize findings from the schistosomiasis model, which illustrate opposing activities for IL-13 and IL-13R alpha 2 in health and disease.