Dataset Information

Right atrial cavernous hemangioma.

ABSTRACT: Benign cardiac hemangiomas are rare tumors that may present in or out side of the heart, epicardium being the most common site. Echocardiography is the method of choice in diagnosing cardiac masses and though 3D TEE may seem to add exact information about the location, the time constraint in doing a comprehensive examination along with 3D rendering inside operation room may become an hindrance.

SUBMITTER: Shashikanth M

PROVIDER: S-EPMC7559951 | biostudies-literature | 2020 Jul-Sep

REPOSITORIES: biostudies-literature

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Right atrial cavernous hemangioma.

Shashikanth Manikappa M Nicola Sandler S Yi Chen C Julian Smith S

Annals of cardiac anaesthesia 20200701 3

Benign cardiac hemangiomas are rare tumors that may present in or out side of the heart, epicardium being the most common site. Echocardiography is the method of choice in diagnosing cardiac masses and though 3D TEE may seem to add exact information about the location, the time constraint in doing a comprehensive examination along with 3D rendering inside operation room may become an hindrance. ...[more]

PMID: 32687093

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Project description:BackgroundCavernous hemangiomas, more accurately defined as cavernous venous malformations, constitute the most common primary intraorbital tumors of adults comprising 4-9% of all tumors,[4] and the second most frequent cause of unilateral proptosis after thyroid-related orbitopathy.[3] Over 80% are located within the intraconal compartment, most commonly in the lateral aspect.[1] Surgical treatment for orbital cavernous hemangioma is generally required in symptomatic cases, optic nerve compression, and cosmetically disfiguring proptosis.[2] Transcranial approaches, the most familiar approaches for neurosurgeons, provide wide access to the entire superior and lateral orbit. They usually offer direct visualization, allowing for a safer dissection, while minimizing significant injury to the native neural and vascular anatomy of the orbit.[5] Although transcranial approaches continue to evolve, in many cases, they have been supplanted by endoscopic skull base approaches and modifications to deep lateral orbitotomy approaches.[5].Case descriptionA 62-year-old male patient presented with slowly expanding left proptosis, which he had first noticed 3 years before presentation. He was already blind in his right eye due to a history of traumatic amaurosis in childhood. The left eye examination revealed severe proptosis with restricted eye movement in all directions and significant visual impairment (visual acuity of 20/300, expressed by Snellen test, with no improvement on correction). MRI of the orbit showed a large left superolateral intraconal cavernous hemangioma compressing and displacing the optic nerve, with the typical feature of slow gradual irregular enhancement with delayed washout on contrast-enhanced image. A one-piece modified orbitozygomatic approach was performed and a total en block resection was achieved. The bone flap was fixed with titanium miniplates and screws, the temporal muscle and the skin were closed in a standard fashion. The patient did not present any new deficit in the postoperative period. The patient had good functional and cosmetic outcomes with resolution of proptosis, restoration of eye movements, and improvement of visual acuity in the 3-month follow-up. Postoperative MRI showed total resection.ConclusionThe orbitozygomatic approach for large orbital cavernous hemangioma provides satisfactory orbital decompression and large working space, reduces traction, and increases visualization and freedom to dissect small vessels and nerves that may be tightly attached to the tumor pseudocapsule.

Project description:A cavernous hemangioma, well-known as vascular malformation, is present at birth, grows proportionately with the child, and does not undergo regression. Although a cavernous hemangioma has well-defined histopathological characteristics, its origin and formation remain unknown. In the present study, we characterized the cellular heterogeneity of cavernous hemangioma using single-cell RNA sequencing (scRNA-seq). The main contribution of the present study is the discovery of mesenchymal stem cells (MSCs) that cause cavernous hemangioma formation and may be abnormally or incompletely differentiated from epiblast stem cells. EGFR inhibitors are the potential drugs to treat cavernous hemangiomas. Other new findings include the responsive ACKR1 positive endothelial cell (ACKR1+EC) and BTNL9 positive endothelial cell (BTNL9+EC) and the BTNL9-caused checkpoint blockade enhanced by the CXCL12-CXCR4 signalling. The activated CD8+T and NK cells may highly express CCL5 for their infiltration in cavernous hemangiomas, independent on the tumor cell-derived CCL5-IFNG-CXCL9 pathway. The plasmacytoid dendritic cells (pDCs) function for anti-tumour as CD8+T cells in cavernous hemangiomas. The oxidised low-density lipoprotein (oxLDL) and scavenger receptors (SRs) may play an important role in the immune responses in the tumour microenvironment of cavernous hemangiomas. Notably, we propose that oxLDL induces the oxLDL-OLR1-NLRP3 pathway in M1-like macrophages via the over-expression of OLR1, whereas oxLDL induces the oxLDL-SRs-C1q pathway in M2-like macrophages via the over-expression of many SRs (LILRB5, etc) except OLR1. The highly expressed EREG in M1-like macrophages and the highly expressed EGFR in MSCs may cause MSC proliferation and the tumour progression. The present study revealed the origin of cavernous hemangiomas and reported the marker genes, cell types and molecular mechanisms, which are associated with the origin, formation, progression, diagnosis or therapy of cavernous hemangiomas. Our discoveries facilitate the development of gold standards for molecular diagnosis and effective drugs for treatment, and enrich the fundamental knowledge in the research field of immune, cancer and even cell biology.

Dataset Information

Right atrial cavernous hemangioma.

Publications

Right atrial cavernous hemangioma.

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