Lymphomatoid Granulomatosis: A Unique Complication of Crohn Disease and Its Treatment in Pediatrics

Destombe, Sylvie; Bouron-DalSoglio, Dorothée; Rougemont, Anne‐Laure; Fournet, J; Ovetchkine, Philippe; Champagne, Josette; Deslandres, Colette

doi:10.1097/mpg.0b013e3181b64586

Cited by 8 publications

(9 citation statements)

References 20 publications

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“…Lymphomatoid granulomatosis is an angioinvasive/angiodestructive, usually oligoclonal or monoclonal proliferation of CD20-positive B cells (some of which are infected with EBV) among a background of polyclonal, reactive CD3-positive T cells (Table 7) (22,23,25,40,(50)(51)(52)(53)(54)(55). This disease was originally described by Liebow, who recognized it as a malignant lymphoproliferative B-cell process with an angiocentric distribution reminiscent of granulomatosis with polyangiitis (formerly Wegener granulomatosis).…”

Section: Lymphomatoid Granulomatosismentioning

confidence: 99%

Primary Pulmonary Lymphoid Lesions: Radiologic and Pathologic Findings

Sirajuddin¹,

Raparia²,

Lewis³

et al. 2016

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The pulmonary lymphoid system is complex and is composed of two compartments: the pulmonary lymphatics and the bronchus-associated lymphoid tissue (BALT). Additional important cells that function in the pulmonary lymphoid system include dendritic cells, Langherhans cells, macrophages, and plasma cells. An appreciation of the normal lymphoid anatomy of the lung as well as its immunology is helpful in understanding the radiologic and pathologic findings of the primary pulmonary lymphoid lesions. Primary lymphoid lesions of the lung arise from the BALT and are uncommon. However, they are increasingly recognized within the growing number of posttransplant patients as well as other patients who are receiving immunosuppressive therapies. Primary lymphoid lesions encompass a wide range of benign and malignant lesions. Benign lymphoid lesions of the lung include reactive lymphoid hyperplasia, follicular bronchiolitis, lymphoid interstitial pneumonia, and nodular lymphoid hyperplasia. Malignant lymphoid lesions of the lung include low-grade B-cell lymphoma of mucosa-associated lymphoid tissue (MALT), other non-Hodgkin lymphomas, and Hodgkin lymphoma. Last, a miscellaneous group of primary lymphoid lesions includes lymphomatoid granulomatosis, posttransplant lymphoproliferative disorders, acquired immunodeficiency syndrome (AIDS)-related lymphoma, and intravascular lymphoma/lymphomatosis. These lesions are best evaluated with multidetector chest computed tomography. The radiologic findings of the primary lymphoid lesions are often nonspecific and are best interpreted in correlation with clinical data and pathologic findings. The purpose of this article is to review pulmonary lymphoid anatomy as well as the most common primary pulmonary lymphoid disorders.

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Section: Lymphomatoid Granulomatosismentioning

confidence: 99%

Primary Pulmonary Lymphoid Lesions: Radiologic and Pathologic Findings

Sirajuddin¹,

Raparia²,

Lewis³

et al. 2016

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103

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“…A patient with autoimmune hepatitis was on 50 mg/day azathioprine for 10 months when she developed fatal LG involving the CNS 6. Prior reports have also described LG associated with 6-MP in a paediatric patient with Crohn's disease 7. A study at the Mayo Clinic demonstrated a significant association between EBV-positive lymphoma and prior treatment with azathioprine/6-MP in patients with IBD, and estimated an incidence of 0.5% 8…”

Section: Discussionmentioning

confidence: 97%

Lymphomatoid granulomatosis associated with azathioprine use for immune-mediated neuropathy

et al. 2016

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Lymphomatoid granulomatosis (LG) is a rare Epstein-Barr virus-driven lymphoproliferative disorder that generally arises in immunosuppressed patients and which can be life-threatening. Here we describe the development of pulmonary LG in a patient on long-term azathioprine for immune-mediated neuropathy. Although azathioprine carries a boxed warning for malignancy, its association specifically with LG, an otherwise rare entity, is poorly recognised. Early recognition of drug-induced LG is critical, since discontinuation of the offending agent, and implementation of effective therapy can provide rapid clinical benefit in some patients. In this case, rituximab was used as an effective treatment for LG, which also provided an additional benefit of controlling the patient's underlying neuropathy. Further research is needed to identify vulnerable patients who are at high risk of developing drug-induced LG.

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“…A review of the literature showed four reported cases. The previously reported individuals with IBD were noted to be in patients with Crohn's disease, all of whom were maintained on 6-Mercaptopurine and one was maintained on Azathioprine [2][3][4][5]. Abdominal pain associated with B symptoms and respiratory symptoms was the common presentation.…”

Section: Discussionmentioning

confidence: 99%

A Rare Case of Lymphomatoid Granulomatosis Occurring with Ulcerative Colitis

Nassereddine¹,

Rafei²,

Tabbara³

et al. 2017

JCPCR

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Lymphomatoid granulomatosis (LyG) is a rare type of angiocentric and angiodestructive lymphoproliferative disorder. We report a rare presentation of lymphomatoid granulomatosis in a patient with ulcerative colitis who's been maintained on 6-Mercaptopurine for prolonged period of time. Although it is not clear whether inflammatory bowel disease (IBD) by itself is associated with such disorder or whether the use of immunosuppressive agents for the treatment of IBD induces EBV related lymphoproliferative disorder, it is important that clinicians are aware of the possible occurrence to ensure appropriate diagnostic evaluation and intervention.

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Lymphomatoid Granulomatosis: A Unique Complication of Crohn Disease and Its Treatment in Pediatrics

Cited by 8 publications

References 20 publications

Primary Pulmonary Lymphoid Lesions: Radiologic and Pathologic Findings

Primary Pulmonary Lymphoid Lesions: Radiologic and Pathologic Findings

Lymphomatoid granulomatosis associated with azathioprine use for immune-mediated neuropathy

A Rare Case of Lymphomatoid Granulomatosis Occurring with Ulcerative Colitis

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