Unusual intracardiac tumor in a child: <i>Inflammatory pseudotumor or “granulomatous” variant of myxoma?</i>

González-Crussí, F.; Vanderbilt, Burton L.; Miller, Jon K.

doi:10.1002/cncr.2820360939

Cited by 46 publications

(14 citation statements)

References 23 publications

(1 reference statement)

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“…Although it has been described in various lesions, cardiac lesions are much more uncommon. With our best knowledge, less than 50 cases of cardiac IMTs have been published in English since the initial description made by Gonzalez-Crussi in 1975 [2]. Owing to its extremely rarity, the etiology of IMT still remains unresolved and the immunologic and infectious postulates are still to be validated.…”

Section: Discussionmentioning

confidence: 99%

A rare cardiac inflammatory myofibroblastic tumor involving aortic valve

Luo

Teng

2017

J Cardiothorac Surg

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BackgroundCardiac inflammatory myofibroblastic tumor (IMT) is an extremely rare benign entity which constitutes a small proportion of primary cardiac tumor.Case presentationWe present a rare case of a 55-year-old symptomatic male patient with a rare cardiac IMT causing left ventricular outflow tract (LVOT) obstruction. The patient received complete tumor resection immediately with uneventful postoperative hospital course.ConclusionTo our best knowledge, cardiac IMT involving aortic valve in older adult causing LVOT obstruction has never been reported before. Additionally, we make a literature review on IMT, focusing on the various clinical presentation of this unpredictable tumor.

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Section: Discussionmentioning

confidence: 99%

A rare cardiac inflammatory myofibroblastic tumor involving aortic valve

Luo

Teng

2017

J Cardiothorac Surg

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“…Cardiac IPT/IMT is rare, and only isolated cases have been reported: 39 cases have been reported in the English language literature, and 37 are listed in Table 2 (the two unlisted cases lacked precise data) 3,6,10–33 . The patients ranged in age from 5 weeks to 72 years, with a predilection for females.…”

Section: Discussionmentioning

confidence: 99%

Inflammatory pseudotumor (inflammatory myofibroblastic tumor) of the mitral valve of the heart

Obikane

Ariizumi

Yutani

et al. 2010

Pathology International

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We report a rare case of inflammatory pseudotumor/inflammatory myofibroblastic tumor (IPT/IMT) of the heart, involving the mitral valve. A 58-year-old woman presenting with dyspnea was immediately admitted to the hospital, and found to have congestive heart failure due to the invagination of a tumor-like mass of the mitral valve. This mass arose from and involved almost the entire posterior leaflet of the mitral valve and occupied almost the entire mitral valve orifice. The tumor was a yellowish-white well-circumscribed mass with a smooth surface. The excised mass was 3.0 x 2.3 x 1.8 cm, and consisted of abundant Sudan III-positive foam cells, histiocytes, lymphocytes, plasma cells, and loosely arrayed spindle cells, in vascular-rich fibrous tissue. Immunohistochemistry showed that the spindle cells were positive for vimentin and alpha-smooth muscle actin, and negative for desmin, CD34, and human muscle actin (HHF-35), suggesting they were myofibroblastic cells. The plasma cells and lymphocytes showed no monoclonality. There were few mitotic cells, and, except for the lymphocytes, few Ki-67-positive cells. The findings suggested IPT/IMT. The 39 cardiac IPT/IMT cases appearing in the English-language literature are discussed.

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“…The scarcity of these two conditions: BD is a rare vasculitis with a prevalence which does not exceed 8 to 37/10,000 inhabitants [27,28]. Cardiac NSIPTs are also exceptional [29][30][31], and since their first description in 1975 by Gonzalez-Crussi F [32], fewer than 30 cases have been reported [31]. Thus, and given the extreme rarity of simultaneously having these two conditions "as a mere coincidence" in the general population, the introduced is the only guarantee of a good prognosis.…”

Section: Introductionmentioning

confidence: 99%

Cardiac Inflammatory Pseudotumors in Behçet’s Disease

Bouomrani

Belgacem

2019

JHC

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Introduction Cardiac non-specific inflammatory pseudotumors (NSIPT) are exceptionally associated to Behçet's disease (BD) and represent a real diagnostic and therapeutic challenge. The meaning and the mechanisms of this association are not yet well understood. The purpose of this paper is to study the epidemiologic, therapeutic, and evolutionary characteristics of cardiac NSIPT during BD Methods Systemic review of all reported cases of cardiac NSIPT associated with BD. Results We found only 6 cases of NSIPT associated with BD. Of these six patients, 4 were men (66.66%) and 2 were women (33.33%): Sex ratio =2. The average age was 26.66 years (9-35 years). The pseudotumor was unique in all cases. The chronology of occurrence of these NSIPT compared to the underlying angiitis was variable: inaugural of the disease in 4 cases, and complicating a previously known BD in 2 cases. The surgery was performed in all cases. It was carried out for diagnostic purpose in 4 cases, and therapeutic in the other 2. Additional medical treatment based on systemic corticosteroids with or without immunosuppressants was indicated in 4 patients. The evolution was favorable in 5 cases and a single case was quickly fatal. Recurrence of NSIPT was reported in one patient (20%). Conclusion The results of this review suggest a very likely association between BD and cardiac NSIPT; especially because of the scarcity of these two conditions in the general population, and the epidemiological characteristics clearly different from those of cardiac NSIPTs in the general population. The pathogenic mechanisms common to these two conditions (immune, inflammatory, reactive, and vascular) reinforce this causal link. The main differential diagnoses of these pseudotumors during BD remain cancer and intracardiac thrombosis.

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Unusual intracardiac tumor in a child: Inflammatory pseudotumor or “granulomatous” variant of myxoma?

Cited by 46 publications

References 23 publications

A rare cardiac inflammatory myofibroblastic tumor involving aortic valve

A rare cardiac inflammatory myofibroblastic tumor involving aortic valve

Inflammatory pseudotumor (inflammatory myofibroblastic tumor) of the mitral valve of the heart

Cardiac Inflammatory Pseudotumors in Behçet’s Disease

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