Kasabach-Merritt Phenomenon: A Retrospective Study of Treatment with Vincristine

Haisley-Royster, Camille; Enjolras, O; Frieden, Ilona J.; Garzon, María C.; Lee, Margaret; Laat, P.C.J. de; Madern, Gerard C.; González, Fernández; Frangoul, Hayder; Moine, Philippe; Prose, Neil S.; Adams, Denise M.

doi:10.1097/00043426-200208000-00010

Cited by 211 publications

(159 citation statements)

References 17 publications

(20 reference statements)

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“…Toxicities associated with myelosuppression and neurotoxicity require relevant precautions. In particular, vincristine has been reported to cause loss of deep tendon reflexes, peripheral neuropathy and abdominal autonomic disturbance, such as constipation (21). The effects of angiography and embolization have been shown to be temporary and are used primarily as an adjunct to surgery, in order to minimize bleeding during a planned resection (22).…”

Section: Kms Was First Noted By Kasabach and Merritt In 1940 (1) Enjmentioning

confidence: 99%

Multimodal treatment of Kasabach-Merritt syndrome arising from tufted angioma: A case report

Jiang

Zhao

2017

Oncology Letters

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Abstract. Kasabach-Merritt syndrome (KMS) is a rare type of vascular tumor associated with a severely decreased platelet count. No standard guidelines for the treatment of the disease have been established so far. In the present study, a 1-year-old pediatric patient with KMS arising from tufted angioma was successfully and variously treated with steroids, vincristine, surgery and propranolol for 18 months. Systemic steroids stabilized the platelet count stable, while vincristine reduced the size of the tumor. Due to unpredictable response, the patient was operated. Combination of vincristine and propranolol was introduced post-surgery to improve the severely low platelet count of the patient. Following multimodal therapy for 18 months, there has been no evidence of recurrence or metastasis during 2 years of follow-up. Currently, the patient is alive and well. The management of KMS presents a challenge, and well-designed studies are required to clearly determine the benefits and risks of multidisciplinary treatment.

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Section: Kms Was First Noted By Kasabach and Merritt In 1940 (1) Enjmentioning

confidence: 99%

Multimodal treatment of Kasabach-Merritt syndrome arising from tufted angioma: A case report

Jiang

Zhao

2017

Oncology Letters

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“…Kasabach-Merritt syndrome (K-MS; large hemangioma with thrombocytopenia syndrome) is a serious and life-threatening hemangioma (3)(4)(5)(6)(7), which is often associated with comorbid conditions such as hemangioendothelioma (8)(9)(10) or plexiform hemangioma (tufted angiomas) (11). In addition to the rapid increase in the size of the tumor, other features of the syndrome include the development of a low platelet count, microvascular disease, anemia and blood coagulation dysfunctions (12). In the present study, it was generally considered that vasculogenesis and angiogenesis are involved in the formation of new blood vessels in the hemangioma (13).…”

Section: Introductionmentioning

confidence: 74%

Artesunate inhibits proliferation and invasion of mouse hemangioendothelioma cells in?vitro and of tumor growth in?vivo

et al. 2017

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Abstract. Artesunate has been demonstrated to be a novel potential antitumor agent in numerous studies. However, its efficacy in infantile hemangioma is unknown. The aim of the present study was to investigate the role of artesunate in the control of vascular tumor biological behavior and molecular mechanism using mouse hemangioendothelioma endothelial (EOMA) cells and a nude mouse model. Cell viability, apoptosis and invasion were determined by an MTT assay, flow cytometric analysis and Transwell invasion assay, respectively. Reverse transcription-quantitative polymerase chain reaction and western blotting were utilized to examine the expression of genes and proteins. Inoculated EOMA cells were injected into the subcutaneous tissues of nude mice to observe the effect of artesunate therapy on the vascular tumor, an effect that was similar to that of pingyangmycin (PYM). It was identified that artesunate treatment (0-600 µg/ml) inhibited cell growth in a time-and dose-dependent manner. Artesunate at 300 µg/ml significantly reduced the proliferation and invasion of EOMA cells, and significantly decreased the expression of vascular endothelial growth factor (VEGF)-A, VEGFR-1, VEGFR-2 and hypoxia inducible factor-1α over time; caspase-3 was simultaneously upregulated in vitro. Artesunate significantly inhibited tumor growth, and the curative effect was similar to that observed with PYM in vivo. It was concluded that artesunate could effectively inhibit the growth of vascular tumors, and thus could be a novel drug candidate for the treatment of infantile hemangioma.

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“…[11,[13][14][15]. Only a few previous studies have shown the benefits of adjuvant chemotherapy, and the results were highly estimated.…”

Section: Discussionmentioning

confidence: 99%

Soft Tissue Hemangioendothelioma in Children

Youn

Jung

Kim

et al. 2017

J Korean Assoc Pediatr Surg

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Purpose: Soft tissue hemangioendothelioma (STHE) is a rare vascular tumor, which has a similar prognosis to borderline malignancy. The disease is poorly understood in pediatric cases because of its low incidence; therefore, we investigated treatment strategies for STHE in children. Methods: We retrospectively analyzed 8 patients with STHE, who were pathologically confirmed between January 1995 and June 2015. The median duration of follow-up was 72 months. Results: Five were male and the median age at the time of surgery was 1.2 years. Six patients presented with a palpable mass, and 2 patients experienced facial paralysis. The median tumor size was 4.0 cm. The following tumor locations were observed head (2 patients), neck (2 patients), chest wall (1 patient), sacrococcyx (1 patient), upper limb (1 patient), and lower limb (1 patient). The patients underwent either microscopic complete resection (R0) (3 patients), macroscopic complete resection (R1) (2 patients), or macroscopic incomplete resection (R2) (3 patients). After histopathological examination, 6 patients were diagnosed with kaposiform hemangioendothelioma (HE), one with retiform HE, and one with epithelioid HE. Postoperative sequelae occurred as gait disturbance, hearing impairment, and vocal cord palsy. Tumor recurrence or regrowth occurred in 4 patients. These patients underwent reoperation and IFN therapy; however, in the patient with epithelioid HE, metastasis to the scalp occurred after these therapies. The patient with the head tumor who underwent R2 resection, underwent resection three more times, but died 11 months after the first surgery. Conclusion: When treating STHE in children, R0 resection should be first considered, but recurrence and metastasis should be monitored depending on the size, pathology, and location of the lesion. When major sequelae are expected, function-preserving surgery could be considered, depending on tumor location, size, and nearby organs.

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Kasabach-Merritt Phenomenon: A Retrospective Study of Treatment with Vincristine

Abstract: Vincristine presents a safe and sometimes effective treatment option in the management of KMP.

Cited by 211 publications

References 17 publications

Multimodal treatment of Kasabach-Merritt syndrome arising from tufted angioma: A case report

Multimodal treatment of Kasabach-Merritt syndrome arising from tufted angioma: A case report

Artesunate inhibits proliferation and invasion of mouse hemangioendothelioma cells in?vitro and of tumor growth in?vivo

Soft Tissue Hemangioendothelioma in Children

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