Pulmonary hyalinizing granuloma: a multicenter study of 5 new cases and review of the 135 cases of the literature

被引:29
作者
Lhote, Raphael [1 ]
Haroche, Julien [1 ,2 ]
Duron, Loic [1 ]
Girard, Nicolas [3 ]
Lafourcade, Marie Pierre [4 ]
Martin, Michel [4 ]
Begueret, Hugues [5 ]
Taytard, Andre [6 ]
Capron, Frederique [2 ,7 ]
Grenier, Philippe [2 ,8 ]
Piette, Jean Charles [1 ,2 ]
Cohen-Aubart, Fleur [1 ,2 ]
Amoura, Zahir [1 ,2 ]
机构
[1] Grp Hosp Pitie Salpetriere, AP HP, Ctr Natl Reference Malad Autoimmunes Syst Rares, Serv Med Interne 2, 47-83 Blvd Hop, F-75651 Paris 13, France
[2] Sorbonnes Univ, Univ Pierre & Marie Curie, F-75013 Paris, France
[3] Hop Louis Pradel, Serv Pneumol, Ctr Natl Reference Malad Pulmonaires Rares, Ctr Expert Natl Associe Reseau RYTHMIC Tumeurs Th, Lyon, France
[4] Ctr Hosp Angoulme, Serv Pneumol, Angouleme, France
[5] Ctr Hosp Pessac, Serv Anatomopathol, Pessac, France
[6] Ctr Hosp Pessac, Serv Pneumol, Pessac, France
[7] Grp Hosp Pitie Salpetriere, AP HP, Serv Anatomopathol, Paris, France
[8] Grp Hosp Pitie Salpetriere, AP HP, Serv Radiol, Paris, France
关键词
Hyalinizing granuloma; Lung nodule; 18FDG PET scan; RETROPERITONEAL FIBROSIS; RARE CAUSE; ASSOCIATION; LYMPHOMA; PATIENT; LUNG; PHG;
D O I
10.1007/s12026-016-8852-4
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
Pulmonary hyalinizing granuloma (PHG) is a rare disease characterized by single or multiple benign lung nodules mimicking lung neoplasma. Histologic analysis reveals homogenous hyaline lamellae, usually surrounded by collection of plasma cells, lymphocytes and histiocytes in a perivascular distribution. The clinical and radiological findings have been described in small series, but the long-term outcomes have rarely been reported. The objectives were to describe the clinical, radiological and outcomes of PHG in new cases and through a literature review. Patients with PHG were found by a multicenter search among French departments of internal medicine, pulmonology and anatomo-pathology. Review of the literature was made through the National Library of Medicine's MEDLINE database using keywords "hyalinizing granuloma." Five news cases and 135 cases of the literature were found. There were 82 men and 57 women, mean age at the diagnosis 44.6 years (15-83). Patients were frequently asymptomatic (n = 39, 27.4 %). The nodule was unique in 37 cases (28.9 %) and multiple in 91 cases (71.1 %). 18FDG PET scan revealed hypermetabolism of the nodule in 9/15 cases (60 %). A systemic disease was associated in 65 cases (mainly mediastinal and retroperitoneal fibrosis, autoimmune, tumoral or infectious disease or thromboembolism). The outcomes were evaluated in 73 patients when follow-up was available: 14 patients had a surgical resection of the nodule. Forty-five patients did not receive any immunosuppressive drug. Among these patients, 2 improved, 29 were stable and 14 worsened. Corticosteroids were used as a monotherapy in 19 patients and led to radiological improvement in 8 cases, stabilization in 8 cases and worsening in 3 cases. Five patients were treated with corticosteroids and at least one immunosuppressive drug and 4 patients improved. PHG is a rare benign disease, mimicking lung neoplasma, frequently associated with systemic diseases.
引用
收藏
页码:375 / 385
页数:11
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