Myocardial Inflammatory Pseudotumor and Multiple Thromboses as a Manifestation of Behcet Disease

Leitão, Bruno; Machado, Fabiana De Castro; Soares, Filomena Valente; Souza, Helga Cecília Muniz de; Queiroz, A C C; Santiago, Mittermayer Barreto

doi:10.1097/rhu.0b013e3181b082a0

Cited by 5 publications

(7 citation statements)

References 14 publications

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“…This report well illustrates a common clinical situation of delayed diagnosis of IgG4-RD. Many reports of inflammatory pseudotumor in different sites may be examples of undiagnosed IgG4-RD [ 13 , 14 ]. In the present case, this condition should have been suspected after the histopathological results indicating “chronic inflammatory processes” in the tissue specimens.…”

Section: Discussionmentioning

confidence: 99%

IgG4-Related Disease: A Multispecialty Condition

Santana¹,

Fonseca²,

Santiago³

2014

Case Reports in Rheumatology

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IgG4-related disease (IgG4-RD) is a recently recognized group of conditions, characterized by tumor-like swelling of involved organs, lymphoplasmacytic infiltrate rich in IgG4-positive plasma cells, variable degrees of fibrosis, and elevated serum IgG4 concentrations. Currently IgG4-RD is recognized as a systemic condition that can affect several organs and tissues. Herein we report the case of a 34-year-old male patient who was admitted to our hospital with diffuse abdominal pain, weight loss, and painful stiffness in his neck. He had a history of tumoral mass of the left maxillary region, right palpebral ptosis with protrusion of the eyeball, and chronic dry cough for about 6 years. Laboratory tests revealed polyclonal hypergammaglobulinemia and increased serum IgG4 levels. Immunohistochemical staining of the maxillary biopsy was compatible with IgG4-RD. He had an excellent response to corticosteroid therapy. This case highlights that IgG4-RD should be included in the differential diagnosis with multisystem diseases.

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Section: Discussionmentioning

confidence: 99%

IgG4-Related Disease: A Multispecialty Condition

Santana¹,

Fonseca²,

Santiago³

2014

Case Reports in Rheumatology

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“…Can et al reported a 12‐year‐old Behçet's disease patient with secondary pseudotumor syndrome due to cerebral vein thrombosis. There was a case of a patient with Behçet's disease with small bowel pathology 6–9 …”

Section: Discussionmentioning

confidence: 99%

“…There was a case of a patient with Behçet's disease with small bowel pathology. [6][7][8][9] In cytogenetic analysis, one third to one half of inflammatory pseudotumor patients present with a genetic translocation on chromosome 2p23, the anaplastic lymphoma kinase (ALK) receptor gene that is often fused with tropomyosin genes. The rate of ALK positivity in cardiac inflammatory pseudotumor is unknown.…”

Section: Discussionmentioning

confidence: 99%

Inflammatory Pseudotumor of the Right Ventricle in a 35‐Year‐Old Woman with Behçet's Disease: A Case Report

Yao

Liu²,

Zhang³

et al. 2012

Echocardiography

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Inflammatory pseudotumor is a rare benign neoplasm. It is common in children and has been reported in various locations throughout the body but rarely in the heart. Behçet's disease is a multisystemic, recurrent, inflammatory disorder. We report a 35-year-old Behçet's disease patient with pseudotumor of the right ventricle and multiple pulmonary emboli. Transthoracic echocardiography showed dilation of right atrium, right ventricle, and a mass in the right ventricle. Multislice computed tomography revealed a large, poorly defined mass infiltrating the groove and multiple pulmonary emboli. Surgery was performed with diagnostic and therapeutic intent. Following sternotomy, we identified a mass in the anterior wall of right ventricle, the outflow tract, and the inflow tract of right ventricle. The histopathologic analysis identified an inflammatory infiltrate composed of lymphocytes, plasma cells, and other inflammatory cells, without mitosis. And there were also some thrombus on top of the mass.

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“…The majority of tumors are firm in consistency, with a few showing mucoid and, rarely, friable areas. Areas of cystic degeneration, Eilers et al 557 20 18 yrs M Constitutional, other IVS, RV, RVOT 3.5 IMT SR 14 21 11 mo M Cardiac RVOT, IVS, PV 2.5 IMT SR 15 22 22 yrs M Constitutional, GI IVS 8.6 IP SR 16 23 49 yrs M Cardiac, respiratory LA, MV, Pveins U IMT SR 17 24 62 yrs F Respiratory, cardiac AV, MV U IP SR 18 25 3 mo F Constitutional, cardiac LV 5.4 IMT T 19 14 5 wks M Respiratory RA 6 IMT U 20 14 2 yrs F Neurologic LV, MV 4 IMT SR 21 14 5 yrs F Neurologic LV, MV 3 IMT U 22 14 10 yrs M Cardiac RVOT 1.5 IMT U 23 14 12 yrs M Neurologic RV 6 IMT U 24 14 17 yrs F Cardiac RV, TV 5 IMT U 25 14 19 yrs F Constitutional LV 3.2 IMT SR 26 14 21 yrs F Constitutional, respiratory IVS, PV, RVOT 4 IMT SR 27 26 69 yrs F Constitutional, respiratory AV, IVS, MV 3 IMT SR 28 27 7 yrs F Cardiac TV, RVOT, PA, PV 5 IMT SR 29 28 54 yrs F A/I AV, AVS, CA, MV, TV 4.8 IP S 30 29 48 yrs F Constitutional AR, AV, PA, RVOT U IMT A, S 31 30 2 mo M Neurologic, cardiac, respiratory RV, RA, TV 2 IMT SR 32 31 2 mo F Cardiac RA 2.5 IMT SR 33 32, 33 35 yrs M Constitutional, respiratory IAS, CS, RA, RV, TV 5 PFT SR 34 34 27 yrs M Cardiac, constitutional, respiratory IVS, RV, LV, P 2.5 IP IB 35 35 13 41 13 wks M U RV 2.5 IMT SR 43 41 1 yrs M U RV 2 IMT SR 44 42 62 yrs M Syncope IAS, RA, AV 5 IP SR 45 43 3 mo F Respiratory IVS U IMT SR 46 44 5 mo F Cardiac IVS, RVOT, PV, PA 3 IMT SR 47 45 4 mo M A/I RA 5 IP SR 48 46 15 yrs M Cardiac, sudden death CA (also spleen by autopsy) U IPT N/A 49 46 5 mo F Respiratory RA, SVC, AVG 2.5 IPT SR 50 47 1 yrs 48 17 yrs M Constitutional, other IAS, IVS, RA, RV, TV U IPT SR 54 …”

Section: Gross Appearancementioning

confidence: 99%

Cardiac Inflammatory Myofibroblastic Tumor

Eilers

Nazarullah

Shipper

et al. 2014

World J Pediatr Congenit Heart Surg

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Although inflammatory myofibroblastic tumors (IMTs) can be found in virtually every major organ, cardiac origin is rare. After recently providing care to a child who presented with a significant myocardial infarction, interest in this rare tumor was piqued. We describe a comprehensive review of cardiac IMT, including information on nomenclature, epidemiology, clinical features, pathogenesis, gross/histological features, immunohistochemical profile, diagnosis, treatment, and prognosis. Fifty-seven cases were identified in the literature. Interestingly, our case represents the seventh case of coronary artery involvement reported. Moreover, it was found that an initial presentation of sudden death most commonly involves the coronary arteries.

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Myocardial Inflammatory Pseudotumor and Multiple Thromboses as a Manifestation of Behcet Disease

Cited by 5 publications

References 14 publications

IgG4-Related Disease: A Multispecialty Condition

IgG4-Related Disease: A Multispecialty Condition

Inflammatory Pseudotumor of the Right Ventricle in a 35‐Year‐Old Woman with Behçet's Disease: A Case Report

Cardiac Inflammatory Myofibroblastic Tumor

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