Analysis of angiographic characteristics of kaposiform hemangioendothelioma and investigation of the value of transcatheter arterial embolization therapy

Tan, Xiaoyun; Liu, Zhenyin; Zhou, Shaoyi; Chen, Kunshan; Zhang, Ming; Xià, Jì; Guo, Yiqun; Zhou, Zijun

doi:10.21037/tp-21-486

Transl Pediatr

2021

DOI: 10.21037/tp-21-486

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Analysis of angiographic characteristics of kaposiform hemangioendothelioma and investigation of the value of transcatheter arterial embolization therapy

Xiaoyun Tan¹,

Zhenyin Liu²,

Shaoyi Zhou³

et al.

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Cited by 2 publications

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“…Due to tumor rarity, delayed diagnosis is frequent [1]. The lesions should be preoperatively distinguished from benign lesions, such as infantile hemangioma because imaging characteristics are different [3].…”

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confidence: 99%

“…The endovascular treatment of vascular spinal lesions has potential to reduce the intraoperative bleeding, shrink the lesion, and avoid mass effect [4, 5]. However, the feeding arteries of KHE are usually numerous and thin, thus embolization may be difficult [3]. The targeted therapy with the mTOR inhibitor (such as sirolimus and everolimus) may have better results than traditional vincristine, and adjunct embolization seems to induce faster resolution of Kasabach-Merritt syndrome [1, 5].…”

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Spinal Kaposiform Hemangioendotheliomas: Is There a Role for Endovascular Treatment?

et al. 2022

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Kaposiform hemangioendotheliomas (KHE) are rare vascular neoplasms composed of groups of capillaries and endothelial cells, forming aneurysms and arteriovenous fistulas. They occur mainly in the extremities during early childhood or infancy, and although they are locally aggressive, the occurrence of metastases is uncommon. A 9-month-old male infant born with a thoracolumbar cutaneous red lesion presented with progressive loss of strength in lower limbs, evolving with progressive flaccid paraplegia and areflexia. The investigation showed extensive intramedullary injury extending from T2 to L2, with associated intradural aneurysms. After an excisional biopsy showing KHE, the intradural lesion was treated endovascularly through transarterial embolization using PVA microspheres2. The uneventful procedure was complemented by corticosteroids and systemic chemotherapy with vincristine, achieving rapid and progressive motor recovery. After 6-months of follow-up, the patient was able to walk with ataxic gait and imaging showed remission of the lesion.KHE generally affect the trunk, retroperitoneum and extremities, but cases involving the head and neck were already described. Complication with Kasabach-Merritt syndrome (KMS) is common, and it is characterized by life-threatening thrombocytopenia, hemolytic anemia, and coagulopathy. Due to tumor rarity, delayed diagnosis is frequent. The lesions should be preoperatively distinguished from benign lesions, such as infantile hemangioma, because imaging characteristics are different. The optimal therapy is still unknown, and although complete surgical excision is considered curative, extensive intramedullary injuries may be difficult to resect due to the risk of hemorrhage. The endovascular treatment of vascular spinal lesions has potential to reduce the intraoperative bleeding, shrink the lesion and avoid mass effect. However, the feeding arteries of KHE are usually numerous and thin, thus embolization may be difficult. The targeted therapy with the mTOR inhibitor (such as sirolimus and everolimus) may have better results than traditional vincristine, and adjunct embolization seems to induce faster resolution of KMS. Endovascular treatment is a valuable tool to improve outcomes and quality of life, therefore multidisciplinary treatment is encouraged.

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Spinal Kaposiform Hemangioendotheliomas: Is There a Role for Endovascular Treatment?

et al. 2022

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Case report: Transcatheter arterial embolization in a newborn with cervical rapidly involuting congenital hemangioma and Kasabach–Merritt phenomenon

Peng

Lin²,

Liu³

et al. 2023

Front. Pediatr.

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Congenital hemangiomas (CHs) are rare vascular tumors and do not exhibit progressive postnatal growth. The incidence is less than 3% of all hemangiomas. Most CHs have a favorable prognosis; however, the Kasabach–Merritt phenomenon (KMP) is a rare but life-threatening complication in CHs that requires aggressive treatment. Medical treatments with corticosteroids and interferon have been suggested. Surgical resection can be considered for the treatment of complicated CHs in medically resistant lesions. Vascular embolization could be an alternative method if surgery is not considered feasible. Herein, we report a case of a 9-day-old newborn who underwent arterial embolization for a CH with KMP, combined with sirolimus treatment, and the outcome was favorable. The hemangioma completely regressed by 3 months and rapidly involuting congenital hemangioma (RICH) was diagnosed. Our successful experience with treating RICH associated with KMP revealed that RICH can have potentially serious complications although they usually resolve rapidly after birth without treatment. Surgical resection is considered to be the standard method for the treatment of medically resistant vascular tumors, but it is difficult to perform during the active phase of KMP due to acute bleeding and severe coagulopathy. Arterial embolization is feasible and can be used as an alternative to surgical resection, even in small babies.

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Analysis of angiographic characteristics of kaposiform hemangioendothelioma and investigation of the value of transcatheter arterial embolization therapy

Cited by 2 publications

References 17 publications

Spinal Kaposiform Hemangioendotheliomas: Is There a Role for Endovascular Treatment?

Spinal Kaposiform Hemangioendotheliomas: Is There a Role for Endovascular Treatment?

Case report: Transcatheter arterial embolization in a newborn with cervical rapidly involuting congenital hemangioma and Kasabach–Merritt phenomenon

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