Luo Cheng,Gao Zhipeng,Dong Zhan,et al.Kaposiform hemangioendothelioma of bone: a case report with a literature review[J].Journal of Clinical Pediatric Surgery,,20():145-152.[doi:10.12260/lcxewkzz.2021.02.008]
Kaposiform hemangioendothelioma of bone: a case report with a literature review
- Keywords:
- Kaposiform Hemangioendothelioma; Bone; Child
- CLC:
- R543 R732.2
- Abstract:
- Objective To explore the clinical manifestations,pathological features,diagnoses,treatments and outcomes of pediatric Kaposiform hemangioendothelioma (KHE) of bone.Methods A case of pediatric KHE of bone was reported along with more cases from the databases of Wanfang,Weipu,CNKI,PubMed and Medline prior to May 2020.Results The lesion of this child was originally derived from calcaneal.After a definite diagnosis through biopsy and pathological examination,treatment of sirolimus was offered at another hospital.After a follow-up period of 66 months,the child survived with disease.A total of 15 pediatric KHE of bone were reported without obvious cutaneous lesion.Only 1 case (1/15,6.5%) with clavicular lesions developed KMP.There were pain (n=11),local swelling (n=5),tenderness (n=5) and restricted movement (n=7).Among 4 children with spinal lesions,there were scoliosis (n=2) and claudication (n=1).Among 9 children receiving radiographic examination,there were lytic lesion (n=7),sclerosis (n=3),uneven bone density (n=2) and thinning of bone cortex (n=1).Among 11 children with CT examination,there were osteolytic changes (n=11) and thinning or destruction of bone cortex (n=7).Among 9 children with MRI examination,there were low T1-weighted & high T2-weighted signal (n=7),high T1-weighted & high T2-weighted signal (n=1) and low T2-weighted signal (n=1).And 3-phase bone scan hinted at higher bone uptake (n=3).Complete treatment data were available for 12 children,the treatments included wide lesional excision (n=6),sirolimus (n=2),vincristine (n=1),thalidomide & celecoxib (n=1),spinal fusion fixation (n=1) and non-treatment (n=1).The mean follow-up period was 45.9(6-186) months.Seven cases survived without any evidence of disease (symptoms/signs disappear,negative imaging results,no tumor recurrence,progression or metastasis) while another 5 cases survived with disease (clinical presentations ease or disappear,imaging results of stable lesions or reduction,no tumor progression or metastasis).The mean follow-up time of 7 survivors without any evidence of disease was 52.9(6-186) months,including surgery (n=6) and vincristine dosing (n=1).The mean follow-up time of 5 survivors with disease was 35.8(8-66) months.The treatments included sirolimus (n=2),thalidomide & celecoxib (n=1),spinal fusion fixation (n=1) and non-treatment (n=1).Conclusion Pediatric KHE of bone is extremely rare and its clinical and imaging presentations are generally non-specific.Early biopsy and pathological examination are helpful for its diagnosis.Children with complete tumor resection have an excellent prognosis,suggesting that surgical resection is a first choice.Drug therapy for pediatric KHE of bone should be standardized and confirmed by prospective multi-center large-sample studies.
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Memo
收稿日期:2020-03-03。
基金项目:南京医科大学科技发展基金（编号：NMUB2018106）；江苏省研究生科研创新计划（编号：JX10413667）
通讯作者:楼跃,Email:jsnjly@126.com