Haemangiomas and vascular malformations of the limb in children

Steven, Mairi; Kumaran, N; Carachi, Robert; Desai, Ashish; Bennet, G.C.

doi:10.1007/s00383-007-1905-y

Cited by 16 publications

(8 citation statements)

References 8 publications

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“…The age of patients with surgically removed vascular malformations varied from 4 wk to 18 y. Localization was in the head and neck region as well as at the extremities, which is in accordance with the literature (8). All vascular malformations Table 2).…”

Section: Resultssupporting

confidence: 82%

β1-Adrenoceptor mRNA level reveals distinctions between infantile hemangioma and vascular malformations

et al. 2013

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Background: Infantile hemangioma (Ih) is the most frequent vascular tumor of early childhood. Recently, propranolol, a nonselective β 1 -and β 2 -adrenoceptor inhibitor, was introduced into the therapy of severe proliferating Ih with excellent results. however, the underlying mechanism of action of propranolol is still unclear. Methods: We performed immunohistochemistry for cluster of differentiation 31 (cD31), D2-40, glucose transporter-1 (GLUT-1), and Ki67 in order to characterize 21 vascular anomalies (nine Ih, seven venous malformations (VMs), and five lymphatic malformations (LMs)). Furthermore, we analyzed the expression of β 1 -, β 2 -, and β 3 -adrenoceptor mRNA in these specimens as well as in hemangioma-derived stem cells by quantitative real-time PcR (qPcR). results: We show that the expression of β 1 -adrenoceptor mRNA is 10.7-fold higher in Ih independent of the proliferative or regressive phase as well as 2.5-fold higher in hemangioma-derived stem cells as compared with β 2 -adrenoceptor mRNA. In LM, the expression of β 2 -adrenoceptor mRNA was ninefold higher than that of β 1 -adrenoceptor mRNA. VM showed low expression levels of all β-adrenoceptor mRNAs, and β 3 -adrenoceptor mRNA was hardly detectable in any specimens examined. conclusion: These results provide the first evidence of distinctions between Ih and vascular malformations with regard to β-adrenoceptor subtype mRNA levels. r ecently, propranolol, a nonselective β-adrenoceptor inhibitor, was introduced into the therapy of severe proliferating infantile hemangioma (IH) (1). Propranolol treatment in IH results in fast growth inhibition and rapid tumor regression within a few days of therapy. This novel medical treatment has not been developed in a classic way; it was accidentally observed during corticosteroid therapy, the so far standard therapy for proliferating IH (2) when cardiac hypertrophy as a side effect is treated by propranolol. The molecular mechanism of propranolol on IH proliferation is unknown at present. A possible method of action could be vasoconstriction or induction of endothelial cell apoptosis (3). Inhibition of β-adrenoceptors has been suggested to have a negative impact on tumor development as well as a direct effect on endothelial cells by downregulation of angiogenesis factors such as vascular endothelial growth factor, matrix metalloproteinase-2, and matrix metalloproteinase-9 (4,5). A possible involvement of the β 2 -adrenoceptor/cyclic adenosine monophosphate pathway has been described in tumor vascularization of glioblastoma (4).IH is the most frequent benign tumor of early childhood that shows specific biological features: after an initial phase of proliferation during the first year of life, a long-lasting period of spontaneous regression over several years is observed (6). The underlying molecular mechanism could not be completely determined because of a lack of models for IH. Until recently, neither IH cell lines nor animal models were available. Endothelial cells from IH show specific expression of glucos...

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Section: Resultssupporting

confidence: 82%

β1-Adrenoceptor mRNA level reveals distinctions between infantile hemangioma and vascular malformations

et al. 2013

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“…Tiny canulae are required in young infants as feeding vessels can be minute. There is a risk of infarction of healthy tissue and reports exist in the literature of stroke (O'Regan et al , ), limb loss and later leg length discrepancy (Steven et al , ). Pain and pyrexia from tissue necrosis may also occur.…”

Section: Mechanical Options and Radiotherapymentioning

confidence: 99%

Recent advances in the pathobiology and management of Kasabach–Merritt phenomenon

et al. 2015

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SummaryKasabach-Merritt Phenomenon (KMP) refers to the clinical constellation of thrombocytopenia, consumptive coagulopathy and purpura associated with Kaposiform haemangioedothelioma or tufted angioma, but not the more common infantile haemangioma. It shows a variable and unpredictable response to traditional pharmacological agents, such as steroids, vincristine or interferon alpha 2a or 2b. More recently, the interaction between platelets and endothelial cells and the proangiogenic phenotype that results has been recognized to underly the pathogenesis of this disorder. Recent efforts have attempted to target the platelet by using antiplatelet agents and by the withholding of platelet transfusions even in those patients who have significant thrombocytopenia and laboratory evidence of coagulopathy. Excellent response rates and prompt results have been achieved by combining antiplatelet therapy with vincristine, without the need for steroid use. This synergistic approach moves away from the conventional wisdom of treating the underlying lesion to control the coagulopathy. Sirolimus, which is directed against the PI3/AKT/ mTOR downstream signalling pathway involved in lymphangiogenesis, has also shown promising results, although further study is needed.

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“…The role of surgery and intervention in current management is still not clear [21]. In case of uncontrollable bleeding of severe diffuse cavernous hemangioma of the lower limbs, combination of superficial femoral artery ligation and supergenual thigh amputation is an effective and safe choice.…”

Section: Discussionmentioning

confidence: 98%

Treatment of severe diffuse cavernous hemangioma of the lower limb by combination of superficial femoral artery ligation and supergenual thigh amputation

Yin

Shang

et al. 2009

Front. Med. China

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Severe diffuse cavernous hemangioma of the lower limb is rarely seen among young people and sometimes can be a fatal challenge for operation. We reported a case of diffuse cavernous hemangioma involving both skin and muscles of the left lower limb and perineal region in an adolescent patient. The patient who had previously undergone a local hemangioma resection of the foot ultimately ended in superficial femoral artery ligation and supergenual thigh amputation of the left upper leg because of uncontrollable massive bleeding of anastomotic stoma.

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Haemangiomas and vascular malformations of the limb in children

Cited by 16 publications

References 8 publications

β1-Adrenoceptor mRNA level reveals distinctions between infantile hemangioma and vascular malformations

β1-Adrenoceptor mRNA level reveals distinctions between infantile hemangioma and vascular malformations

Recent advances in the pathobiology and management of Kasabach–Merritt phenomenon

Treatment of severe diffuse cavernous hemangioma of the lower limb by combination of superficial femoral artery ligation and supergenual thigh amputation

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