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Dual-mode Imaging of Cutaneous Tissue Oxygenation and Vascular Function
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Kaposiform hemangioendothelioma without cutaneous involvement.

Yi Ji1, Siyuan Chen2, Lizhi Li3

  • 1Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China. jijiyuanyuan@163.com.

Journal of Cancer Research and Clinical Oncology
|October 8, 2018
PubMed
Summary

Kaposiform hemangioendothelioma (KHE) without skin lesions can cause severe complications, including death, especially when associated with Kasabach-Merritt phenomenon (KMP). Early diagnosis and aggressive treatment, particularly with sirolimus, are crucial for better outcomes in KHE.

Keywords:
Cutaneous involvementKaposiform hemangioendotheliomaKasabach–Merritt phenomenonMorbidityMortality

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Area of Science:

  • Vascular Anomalies
  • Pediatric Oncology
  • Dermatology

Background:

  • Kaposiform hemangioendothelioma (KHE) is a rare vascular tumor.
  • KHE typically presents with cutaneous involvement, but visceral or deep-seated KHE without skin lesions poses diagnostic and therapeutic challenges.

Purpose of the Study:

  • To characterize the clinical features and management of patients diagnosed with Kaposiform hemangioendothelioma (KHE) lacking cutaneous manifestations.
  • To identify factors associated with poor prognosis and treatment response in this specific KHE subtype.

Main Methods:

  • Retrospective review of electronic patient records from six hospitals in China.
  • Inclusion criteria: patients diagnosed with KHE without cutaneous involvement.
  • Data analysis focused on clinical presentation, complications, management strategies, and outcomes.

Main Results:

  • Thirty patients (56.7% male, mean age 55.6 months) were identified.
  • Kasabach-Merritt phenomenon (KMP) occurred in 46.7% of patients, significantly associated with trunk lesions (OR 10.0).
  • Common complications included severe anemia and decreased range of motion. Sirolimus showed better response compared to other treatments. Mortality was 10%, primarily in KMP patients with specific adverse features.

Conclusions:

  • Kaposiform hemangioendothelioma (KHE) without cutaneous involvement can lead to significant morbidity and mortality.
  • Increased clinical awareness is necessary for early diagnosis and prompt, aggressive management to prevent severe complications.
  • Sirolimus demonstrated efficacy in patients with KHE lacking skin lesions.