Chinese Journal of Clinical Oncology Aug. 2007, Vol. 4, No. 4 P 288~292 Gang Zhou et al. 288 Kaposiform Hemangioendothelioma: Case Report and Literature Review Gang Zhou 1 Shuhua Yang 1 Xiu Nie 2 Jingyuan Du 1 Jin Li 1 Jianxiang Liu 1 Renhao Ze 1 Yu He 1 1 Department of Orthopedics, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430022, China 2 Department of Pathology, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430022, China Correspondence to: Shuhua Yang E-mail: shuhuayabc@vip.sina.com Received April 19, 2007; accepted May 21, 2007. KEYWORS: histopathology; kaposiform hemangioendo- thelioma; bone; vascular tumors. Kaposiform hemangioendothelioma (KHE) is a rare locally ag- gressive vascular tumor that mainly occurs in children and early adolescents. It is often associated with the Kasabach–Meritt phenomenon which is marked by severe thrombocytopenia and a variable degree of anemia. The tumor mostly is located in the superfcial or deep soft tissue mass of the extremities. Herein, we report an unusual case of kaposiform hemangioendothelioma that, for the frst time, was found in the proximal medullary cavity of the tibia and fbula of an 8-year-old boy. This patient was frst di- agnosed as fbrous dysplasia. He underwent wide local excision, but the mcroscopic examination of the mass was confrmed to be kaposiform haemangioendothelioma (KHE) based on morpholog- ical and immunohistochemical fndings, which were characterized by having features common to capillary hemangioma and Kaposi sarcoma. Case Report This case represents the frst report of this disease occurring in the proximal medullary cavity of the tibia and fibula. An otherwise healthy 8-year-old boy felt pain in his left leg. He was not easily bruised or subject to bleeding. The discomfort slowly aggravated over the months. The boy had been born to healthy parents at 40 weeks of gestation by normal delivery. He had no intrauterine dis- ease or perinatal problems. He was vaccinated as scheduled, did not undergo any surgery, and was not admitted for other diseases. On physical examination, the child was afebrile without abnor- mal vital signs or weight loss. No tints of blue, pink, or red on his left leg could be seen and no lymph nodes were palpable. The amount of knee extension was 170 degrees and the amount of knee fexion was 65 degrees, both were limited. Tenderness could be felt by the boy when he was deeply pricked. Laboratory results showed the following: alkaline phosphatase (42, 36~213 U/L), normal blood cell counts, hemoglobin (123 g/L, 120~160 g/L), erythrocytes (4.44×10 12 /L, 4.0~5.5×10 12 /L), leukocytes (9.20× 10 9 /L, 4.0~10×10 9 /L) and platelets (273×10 12 /L, 100~300×10 12 /L). A conventional X-ray examination of the proximal tibia and fbula (Fig.1) showed a large, centrally located, lytic lesion in the me- taphyseal-epiphyseal area with thinning of the surrounding cortex, minimal dilatation and fairly sharp margination that would suggest CJCO http://www.cjco.cn E-mail:cocr@eyou.com Tel(Fax):86-22-2352 2919 [SpringerLink] DOI 10.1007/s11805-007-0289-z