Pulmonary aneurysms and intracardiac thrombi due to Behçet’s disease in an African-American adolescent with oculocutaneous albinism

Endo, L.M.; Rowe, Steven M.; Romp, Robb L.; Buckmaster, Mark A.; Atkinson, T. Prescott

doi:10.1007/s10067-006-0426-8

Cited by 29 publications

(8 citation statements)

References 11 publications

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“…Infliximab may inhibit the activation of infiltrated mononuclear cells by blockade of TNF and TNF-producing cells, resulting in the prevention of thrombosis. [16][17][18][19][20] After four years of infliximab, azathioprine, low dose steroid and oral anticoagulant treatment, our patient had no complaint or new thrombus. Anticoagulation treatment together with immunosuppressive therapy is an advised treatment option in Behçet's cases with intracardiac thrombus formation.…”

Section: Discussionmentioning

confidence: 74%

An adolescent case of extensive Behçet`s disease successfully treated with Infliximab

Isiyel¹,

Bakkaloğlu²,

Oğuz³

et al. 2019

Turk J Pediatr

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Cardiac involvement is an uncommon and life-threatening complication ofBehçet's Disease. We present a 14-year-old boy, admitted to our hospital for recurrent hemoptysis. In his radiologic evaluation, a right ventricular thrombus and pulmonary arterial aneurysm were identified. He was diagnosed with Behçet's Disease, and then he received prednisone and cyclophosphamide. However, his cardiac thrombus enlargened. After his treatment was replaced with infliximab, the pulmonary aneurysms regressed, and the cardiac thrombus disappeared. In conclusion, infliximab should be considered as a reliable option for vascular Behçet's Disease resistant to conventional treatment.

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Section: Discussionmentioning

confidence: 74%

An adolescent case of extensive Behçet`s disease successfully treated with Infliximab

Isiyel¹,

Bakkaloğlu²,

Oğuz³

et al. 2019

Turk J Pediatr

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“…Azathioprine, colchicine and infliximab were the other used drugs. [5][6][7][20][21][22][23][24][25] Despite all combined treatment options, we could not avoid mortality in our case.…”

Section: Discussionmentioning

confidence: 99%

Is Immunosuppressive and Thrombolytic Therapy Really Effective in a Patient With Intracardiac Thrombosis and Pulmonary Artery Aneurysm due to Behçet’s Disease?

2019

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Behçet's disease (BD) is a rare, multisystemic, chronic vasculitic disorder with unknown etiology. The most common signs of BD are recurrent aphthous oral ulcers, genital ulcers, ocular lesions, dermatological lesions, and arthritis. 1,2 BD may also affect the pulmonary and cardiovascular systems. 2-4 Frequency of BD is low but intracardiac thrombus (IcT) formation is even less common; this serious complication has been reported only as case reports. 5-7 Pulmonary artery aneurysm (PAA) is also very rare, and only a few pediatric patients have been reported in the literature. 6,7 IcT accompanying PAA is extremely rare in the pediatric age group. In this article, we present an unusual case of an adolescent patient with PAA and IcT who did not survive despite all medical therapies. CASE REPORT A 16-year-old male patient was admitted to the hospital with complaints of productive cough, fever, and chills. Despite oral amoxicillin/ clavulanic acid treatment, the patient's complaints increased; he developed shortness of breath and was admitted to another hospital. The patient had weight loss of 8 kg in the prior three months. Two years before admission, the patient was suspected to have BD because of recurrent anterior uveitis and treated with infliximab. The patient never developed oral or genital ulcers, arthralgia, or neurological complaints during follow-up. There was no family history of BD or consanguinity. A written informed consent was obtained from the parents of the patient.

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“…Full resolution of thrombosis was reported with intravenous streptokinase plus corticosteroid treatment [12, 23]. A case with multiple ICTs has needed thrombolytic treatment along with anticoagulation, cyclophosphamide, and anti-TNF agent for resolution of ICT [47]. …”

Section: Discussionmentioning

confidence: 99%

Behçet’s Disease and Intracardiac Thrombosis: A Report of Three Cases

Düzgün

Küçükşahin

Atasoy

et al. 2013

Case Reports in Rheumatology

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We present three patients with Behçet's disease associated with intracardiac thrombus and pulmonary vascular involvement. One of these patients had also Budd-Chiari syndrome. All patients were treated with corticosteroid plus monthly intravenous cyclophosphamide as first line treatment and with no recurrences. Immunosuppressive therapy was successful in the treatment of intracardiac thrombus and also in the regression of pulmonary vascular thromboses in these patients. Intracardiac thrombus in Behçet's disease is rarely seen. Behçet's disease should be remembered in the differential diagnosis of the patients with intracardiac mass, especially in patients from the Mediterranean and Middle East populations.

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Pulmonary aneurysms and intracardiac thrombi due to Behçet’s disease in an African-American adolescent with oculocutaneous albinism

Cited by 29 publications

References 11 publications

An adolescent case of extensive Behçet`s disease successfully treated with Infliximab

An adolescent case of extensive Behçet`s disease successfully treated with Infliximab

Is Immunosuppressive and Thrombolytic Therapy Really Effective in a Patient With Intracardiac Thrombosis and Pulmonary Artery Aneurysm due to Behçet’s Disease?

Behçet’s Disease and Intracardiac Thrombosis: A Report of Three Cases

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