Syphilitic Pulmonary Inflammatory Pseudotumor: A Diagnostic Challenge.

A 46-year-old man presented with nonproductive cough and lower limb swelling. Chest radiograph showed a left lower lobe lung mass and multiple subpleural nodules. Other investigations revealed that he had nephrotic syndrome. Core biopsies of the left lower lobe lung mass showed features of inflammatory pseudotumor with endarteritis obliterans and a lymphoplasmacytic infiltrate. Immunohistochemical stain for Treponema pallidum was positive. Resolution of the lung mass and nephrotic syndrome was achieved after treatment with intramuscular benzathine benzylpenicillin. The differential diagnosis of pulmonary inflammatory pseudotumor, manifestations of pulmonary syphilis, and a literature review of secondary syphilis of the lung are discussed.

Pulmonary Mycobacterium Spindle Cell Pseudotumor in Patient With Liver Transplant.

We report a case of liver transplant patient who presented with lung masses, found to be Mycobacterium spindle cell pseudotumors. The masses demonstrated hypermetabolic activities on positron emission tomography. Core biopsy revealed sheets of spindle histiocytic cells with abundant acid-fast bacilli identified as Mycobacterium avium-intracellulare complex. This finding is a rare presentation of Mycobacterium infection, mainly nontuberculous Mycobaterium. It is characterized by a benign, spindle cell mass-forming reaction. Most of the reported cases had acquired immune deficiency syndrome or organ transplant. Histopathology illustrating the proliferation of spindle cell shaped histiocytes containing numerous acid-fast bacilli is the gold standard for diagnosis. The standard treatment has not been well established; previously reported cases followed the standard treatment for Mycobacterium based on organ involvement. Our case is the first case to our knowledge that reports pulmonary Mycobacterium spindle cell pseudotumors in a liver transplant recipient.

Afectación pulmonar bilateral. Opciones no quirúrgicas del pseudotumor inflamatorio, a propósito de un caso / Bilateral Pulmonary Involvement. Non-Surgical Options for Inflammatory Pseudotumor - A Case Report

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Varón con síndrome constitucional y adenopatías mediastínicas. A propósito de un caso / Patient with constitutional syndrome and mediastinal adenopathies. A case report

La sarcoidosis es una enfermedad granulomatosa sistémica de causa desconocida, caracterizada por la presencia de granulomas no caseificantes en los órganos afectados. Afecta típicamente al pulmón de adultos de mediana edad. Se basa en un diagnóstico de exclusión, ya que no existe una prueba diagnóstica definitiva. Como tratamiento de primera línea se recomiendan los corticoesteroides, teniendo como alternativas los inmunosupresores, fundamentalmente el metotrexato, y las terapias biológicas. Presentamos el caso de un paciente de 43 años sin antecedentes de interés, enviado a consultas de neumología por pérdida de peso e imágenes de agrandamiento hiliar bilateral y ensanchamiento mediastínico derecho en la radiografía de tórax

Sarcoidosis is a multisystem granulomatous disorder of unknown etiology, characterized pathologically by the presence of noncaseating granulomas in involved organs (most frecuently the lung of middle aged adults). The most important part of the diagnosis is the exclusion of alternative possibilities, because a definitive diagnostic test for sarcoidosis does not exist. The essence of the treatment are corticosteroids. We present a 43- years-old male patient with no medical history of interest, who was sent to the pneumology outpatient department because of weight loss and bilateral hilar adenopathies and right paratracheal node enlargement on the radiography

Pulmonary inflammatory pseudotumor due to Coxiella burnetii. Case report and literature review.

A 58-year-old man was admitted because of respiratory failure, episodic fever with chilling, cough, malaise, fatigue, myalgia and weight loss lasting for at least one month. Chest x-rays and CT scan of the chest showed bilateral pulmonary consolidations in upper lobes, the left lower lobe, and mediastinal lymphadenopathy. Bronchoscopy with cytology was unremarkable. A needle CT-guided lung biopsy documented an inflammatory pseudotumor, lymphoplasmacytic type. Serology showed high titer antibodies to phase II Coxiella burnetii infection. Therapy with doxycycline and hydroxychloroquine for three months led to a complete resolution of symptoms and radiological findings, and a marked decrease in titers to Q fever.

Prolonged B cell depletion with rituximab is effective in treating refractory pulmonary granulomatous inflammation in granulomatosis with polyangiitis (GPA).

Pulmonary nodule formation is a frequent feature of granulomatosis with polyangiitis (GPA). Traditional induction therapy includes methotrexate or cyclophosphamide, however, pulmonary nodules generally respond slower than vasculitic components of disease. Efficacy of rituximab (RTX) solely for the treatment of pulmonary nodules has not been assessed. In this observational cohort study, we report patient outcomes with RTX in GPA patients with pulmonary nodules who failed to achieve remission following conventional immunosuppression. Patients (n = 5) with persistent pulmonary nodules were identified from our clinic database and retrospectively evaluated. Systemic manifestations, inflammatory markers, disease activity, concurrent immunosuppression, and absolute B cell numbers were recorded pre-RTX and at 6 monthly intervals following treatment. Chest radiographs at each time point were scored by an experienced radiologist, blinded to clinical details. Five patients with GPA and PR3-ANCA were evaluated (2 male, 3 female), mean age 34 (22-52) years. Pulmonary nodules (median 4, range 2-6), with or without cavitation were present in all patients. RTX induced initial B cell depletion (<5 cells/µL) in all patients but re-population was observed in 3 patients. Repeated RTX treatment in these 3 and persistent B cell depletion in the whole cohort was associated with further significant radiological improvement. Radiographic scoring at each time interval showed reduction in both number of nodules (P =  <0.0001) and largest nodule diameter (P =  <0.0001) in all patients for at least 18 months following B cell depletion. In summary, RTX therapy induces resolution of pulmonary granulomatous inflammation in GPA following prolonged B cell depletion.

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.

A rare case of pulmonary cryptococcal inflammatory myofibroblastic tumor diagnosed by fine needle aspiration cytology.

In a recent outbreak in British Columbia (BC), Canada, Cryptococcus gattii, a rare species of Cryptococcus, was noted to affect primarily immunocompetent hosts and cause limited pulmonary or CNS disease. We herein report a rare case of a pulmonary inflammatory myofibroblastic tumor caused by a Cryptococcus infection, presumed to be of the gattii species, in a 20-year-old immunocompetent college student from Vancouver, BC who presented with a large lung mass. The diagnosis was first made on the fine needle aspirate (FNA) material and was confirmed on the concurrent histologic core biopsy. Cryptococcal inflammatory myofibroblastic tumors have been reported, but neither in the lung nor in the setting of an immunocompetent host. Pulmonary cryptococcosis should therefore be considered in the differential diagnosis of a lung mass, even in an immunocompetent host, especially if the clinical history reveals recent travel to British Columbia where Cryptococcus gattii is endemic.

[Inflammatory pseudotumour of the lung as a rare differential diagnosis of a pulmonary mass - a case report and overview of the literature]. / Inflammatorischer Pseudotumor der Lunge als seltene Differenzialdiagnose einer pulmonalen Raumforderung - ein Fallbericht mit Literaturübersicht.

The case of a 37-year-old woman with an inflammatory pseudotumour of the lung is reported. Inflammatory pseudotumour of the lung is a rare disease predominantly occurring in younger patients. Because of its rarity as well as its usually unspecific clinical and radiological presentation, this disease constantly causes diagnostic and therapeutic difficulties. The case report is followed by a short overview of the literature.

Recurrence of inflammatory pseudotumor of the lung after eleven years of remission.

The standard treatment of inflammatory pseudotumor of the lung is surgical excision. However, little data is available on steroid therapy in patients with the unresectable disease. Here, we report a patient with recurrent inflammatory pseudotumor of the lung with pleural involvement who had been successfully treated with corticosteroid eleven years previously. Like the previous treatment, retreatment with corticosteroid proved to be effective for the recurred lesion. In addition, the patient had developed extramammary Paget's disease and bladder cancer after the initial onset of inflammatory pseudotumor. Steroid therapy could be an optional modality in treating unresectable inflammatory pseudotumor, although long-term follow-up is definitely necessary.

[Multicentric inflammatory pseudotumor]. / Seudotumor inflamatorio multicéntrico.

Inflammatory pseudotumor is a non-neoplastic process characterized by irregular growth of inflammatory cells. This type of tumor can arise in any part of the body; most are confined to a single site and are benign. We present the case of a 51-year-old man in whom a solitary pulmonary nodule was detected as an incidental finding. Right pulmonary lobectomy was performed and histopathological analysis led to a diagnosis of inflammatory pseudotumor. The patient subsequently developed tumors in the contralateral lung and cervix, the latter being highly aggressive. We found no reports of this association in the literature, prompting the present case report.

A case of pulmonary inflammatory pseudotumor: Recurrence appearing as several consolidative lesions after complete resection.

Inflammatory pseudotumor (plasma cell granuloma) of the lung is an uncommon nonneoplastic tumor of unknown origin. This tumor typically manifests as a solitary, peripheral, and sharply circumscribed mass. Multiple lesions are seen in about 5% of cases. Resection is recommended for both diagnosis and treatment, and this tumor does not generally recur after complete resection. Here, we report a case of recurrent inflammatory pseudotumor after complete resection; the recurrence was detected as a series of bilateral consolidated lesions with an internal air bronchogram. This is an unusual finding with regard to inflammatory pseudotumors.

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.

Inflammatory pseudotumor of the lung following invasive aspergillosis in a patient with chronic graft-vs.-host disease.

Inflammatory myofibroblastic tumor (IMT) is an uncommon cause of solitary or multifocal lung nodules and can also be rarely found in various other extrapulmonary sites. Although this pseudotumor is benign, it can be locally very aggressive. The pathogenesis of IMT remains unclear; autoimmune or infectious origins have been hypothesized, so far. Here, we report a case of inflammatory pseudotumor of the lung secondary to invasive pulmonary aspergillosis in a patient with chronic graft-vs.-host disease. The 42-year-old patient presented with coughing and hemoptysis as major clinical signs 1 yr after successful HLA-identical stem cell transplantation. Aspergillus fumigatus was cultured from the bronchoscopic lavage, but intensive antifungal treatment could only initially improve the clinical situation. Diagnostic re-evaluation by open-chest biopsy surprisingly revealed an inflammatory pseudotumor responsible for clinical and radiographical deterioration. Both clinical and radiographical signs resolved under long-term steroids and secondary antifungal prophylaxis.

Massive pulmonary pseudotumor.

The term pulmonary pseudotumor may be used to describe a well-demarcated interlobar pleural effusion. Pseudotumors are located within pulmonary fissures and are commonly associated with congestive heart failure and other processes that cause transudative pleural effusions. Pseudotumors are typically diagnosed presumptively on chest radiographs based on their lenticular configuration. We report a case of a massive pseudotumor opacifying one third of the right hemithorax on a frontal radiograph. CT scan of the chest showed a loculated effusion within the oblique fissure measuring 10 x 5 cm. The Hounsfield unit characteristic of the effusion was similar to that of freely layering liquid in the contralateral hemithorax that was shown to be a transudate. The pseudotumor resolved with medical management over 8 weeks. This case proves that even a massive pulmonary pseudotumor will resolve with conservative management.

Bilateral orbital pseudotumor with suprasellar and pulmonary involvement: report of a case.

A 39-year-old man had bilateral proptosis and blurred vision for I week. Computed tomography and magnetic resonance imaging showed signs of bilateral orbital pseudotumor, a suprasellar mass, and pulmonary infiltration. Biopsies from retrobulbar and bronchial sites showed similar inflammatory tissue. His disease resolved with pulsed corticosteroid therapy.

[The anti-inflammatory activity of intal and beta-carotene in a model of experimental granulomatous lung inflammation]. / Issledovanie protivovospalitel'noi aktivnosti intala i beta-karotina na modeli éksperimental'nogo granulematoznogo vospaleniia legkikh.

A-25 sephadex-induced granulomatous inflammation of the lungs in rats was treated with beta-carotene and intal in inhalations. Both drugs showed antiinflammatory activity reducing the area of alveolitis and emphysema in the lungs, number of neutrophils and lymphocytes in the bronchoalveolar fluid. The experimental data allow to recommend further trial of intal and beta-carotene in granulomatous pulmonary diseases.

[Multifocal inflammatory pseudotumor of the lung with good response to corticoids]. / Seudotumor inflamatorio multifocal del pulmón con buena respuesta a corticoides.

A 75-year-old woman was admitted to our hospital with persistent fever, productive cough and systemic symptoms of two months' duration. A chest film showed bilateral infiltration in the form of subpleural plaques with multiple satellite nodules in both lung fields. The biopsy specimens taken by thoracoscopy were compatible with inflammatory pulmonary pseudotumor. Lung resection was ruled out because the lesions were bilateral and corticosteroid treatment was instated. The clinical and radiologic response was excellent and the patient was asymptomatic after 30 months of low-dose corticoid therapy.