Pulmonary hyalinizing granuloma and retroperitoneal fibrosis in an adolescent

Young, Adam S.; Binkovitz, Larry A.; Adler, Brent H.; Nicol, Kathleen; Rennebohm, Robert M.

doi:10.1007/s00247-006-0340-x

Cited by 24 publications

(18 citation statements)

References 9 publications

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“…4 Furthermore, there are a number of older reports in which PHG has been associated with lesions that have now been brought under the ISD umbrella, including sclerosing mediastinitis, retroperitoneal fibrosis, uveitis, and systemic idiopathic fibrosis. [5][6][7][8]22,23 Interestingly, our patient also had pleural fibrosis, which has been described as a thoracic manifestation of ISD. Our case is the first with a demonstration of increased serum and tissue IgG4 to specifically provide the link between PHG and ISD.…”

Section: Discussionmentioning

confidence: 78%

Pulmonary Hyalinizing Granuloma With Associated Elevation in Serum and Tissue IgG4 Occurring in a Patient With a History of Sarcoidosis

Chapman

Gown²,

Mazziotta

et al. 2012

American Journal of Surgical Pathology

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Pulmonary hyalinizing granulomas (PHGs) are unusual fibrosclerotic inflammatory lung lesions. The organ-based manifestations of the recently defined IgG4-related sclerosing disease typically show dense fibrosis and heavy lymphoplasmacytic infiltrates. IgG4-related sclerosing disease is also defined by increased serum IgG4 levels and increased tissue levels of IgG4-positive plasma cells. The morphologic features of PHG overlap with those seen in IgG4-related sclerosing disease, and this suggests that PHG may be a form of IgG4-related sclerosing disease. We present a case of a 51-year-old man with a history of sarcoidosis who presented with slowly enlarging pulmonary nodules. Histologic evaluation of one of the nodules yielded a diagnosis of PHG. Further investigation demonstrated both elevated serum IgG4 and elevated tissue IgG4-positive plasma cells in the PHG. In previous reports, lesions that are now considered part of IgG4-related sclerosing disease were documented in patients also diagnosed with PHG, although these reports date from before the description of IgG4 sclerosing disease. This case provides the first definitive evidence that PHG is part of the spectrum of IgG4-related sclerosing disease.

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Section: Discussionmentioning

confidence: 78%

Pulmonary Hyalinizing Granuloma With Associated Elevation in Serum and Tissue IgG4 Occurring in a Patient With a History of Sarcoidosis

Chapman

Gown²,

Mazziotta

et al. 2012

American Journal of Surgical Pathology

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“…F-18 fluordeoxyglucose positron emission tomography (FDG-PET/CT) can reveal increased metabolic activity in PHG lesions [12]. However, an accurate diagnosis of PHG can only be made with histopathological examination [8].…”

Section: Discussionmentioning

confidence: 99%

“…Some patients with multiple lesions may show progressive enlargement of nodules and increased dyspnea [1, 5]. There is no definitive treatment for multiple nodules, although successful resolution of the nodules with the administration of glucocorticoids has been reported [1, 5, 12, 13]. FDG-PET/CT may demonstrate a decrease in metabolic activity after corticosteroid treatment [12].…”

Section: Discussionmentioning

confidence: 99%

Hyalinizing Granuloma: An Unusual Case of a Pulmonary Mass

Brandão

Marchiori

Zanetti

et al. 2010

Case Reports in Medicine

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We describe the case of pulmonary hyalinizing granuloma in a 34-year-old asymptomatic man who presented with a pulmonary nodule apparent by chest radiography and computed tomography (CT). He had a history of previous treatment for tuberculosis. His laboratory data were normal. Bronchoscopy and CT-guided percutaneous transthoracic fine needle aspiration cytology were inconclusive. The diagnosis was revealed after the histopathological examination of an open lung biopsy.

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“…The PHG can be combined with other diseases, e.g. with retroperitoneal fibrosis, but a pathophysiologic relation between these diseases has not been proven [3,4].…”

Section: Introductionmentioning

confidence: 99%

“…PHG affects mostly adults from the 20's to the 70's showing no sex predilection [6]. The clinical symptoms are typically cough, tiredness, uncharacteristic chest pain, fever, dyspnoea and haemoptysis [1,7] and in 25% of the patients the disease is asymptomatic [4]. There exists no established treatment of PHG.…”

Section: Introductionmentioning

confidence: 99%