Musculoskeletal Tumor Center, Peking University People's Hospital, Beijing, China.
J Pediatr Orthop. 2021 Apr 1;41(4):227-235. doi: 10.1097/BPO.0000000000001738.
Primary benign osseous tumors and tumor-like lesions at the sacrum are rare in the pediatric population and exact surgical strategy is still unclear. In this study, we evaluate the outcome for pediatric patients with benign tumors and tumor-like lesions at the sacrum who were receiving surgical treatment according to our proposed surgical strategy and classification.
We analyzed 49 pediatric patients with sacral benign tumors or tumor-like lesions aged 18 years and below from 2005 to 2018. There were 23 men and 26 women with a mean age of 14.0±3.8 years. Nineteen patients had giant cell tumors (GCTs), 9 aneurysmal bone cysts, 5 osteoblastomas, 5 neurogenic tumors, 3 hemangiomas, 3 teratomas, 2 Langerhans cell histiocytosis, 1 chondroblastoma, 1 fibrous dysplasia, and 1 GCT of tendon sheath. We proposed our surgical plan and surgical classification for pediatric patients with sacral benign tumors or tumor-like lesions.
The mean follow-up duration was 6.2 years (range, 1.0 to 18.9 y). GCTs (39%, 19/49) and primary aneurysmal bone cysts (18%, 9/49) are the top 2 common histologic types. Preoperative selective arterial embolization (SAE) was performed in 12 cases and 24 patients received intraoperative aortic balloon occlusion (ABO) as the preoperative surgical plan. Furthermore, according to tumor location at the sacrum, we classified surgical excision of sacral benign tumors and tumor-like lesions into 3 types. Fourteen cases were classified as type I, 27 as type II, 3 as type III, and 5 patients with neurogenic tumors cannot be classified into this surgical classification. Ten patients had wound complications. Two had femoral artery thrombosis because of ABO application. One had mechanical failure. Rate of local recurrence was 16%. Seven patients with GCTs and 1 with neurogenic tumor had local recurrence. No patient died of disease at the last follow-up. For the assessment of neurological function, the rate of neurological dysfunction was 12% (6/49). Four cases had urinary incontinence, 3 fecal incontinence, and 3 had bowel obstruction. Next, univariate analysis for influence of preoperative SAE and intraoperative ABO on complications demonstrated that both of them exerted no significant influence on the occurrence of oncological and nononcological complications.
The proposed surgical strategy can provide an excellent therapeutic effect for pediatric benign tumors and tumor-like lesions at the sacrum. Preoperative SAE and intraoperative ABO can safeguard pediatric patients with high vascularity of benign tumor at the sacrum during the operation.
Level IV.
原发性良性骨肿瘤和骨肿瘤样病变在儿童中较为少见，确切的手术策略仍不明确。本研究中，我们根据提出的手术策略和分类，评估了接受手术治疗的骶骨良性肿瘤和骨肿瘤样病变患儿的治疗效果。
我们分析了 2005 年至 2018 年间收治的 49 例年龄在 18 岁以下的骶骨良性肿瘤或骨肿瘤样病变患儿的临床资料。其中男 23 例，女 26 例，平均年龄 14.0±3.8 岁。19 例为骨巨细胞瘤（GCT），9 例为动脉瘤样骨囊肿，5 例为骨母细胞瘤，5 例为神经源性肿瘤，3 例为血管瘤，3 例为畸胎瘤，2 例为朗格汉斯细胞组织细胞增生症，1 例为软骨母细胞瘤，1 例为纤维结构不良，1 例为腱鞘 GCT。我们为骶骨良性肿瘤或骨肿瘤样病变患儿提出了手术方案和手术分类。
平均随访时间为 6.2 年（1.0~18.9 年）。GCT（39%，19/49）和原发性动脉瘤样骨囊肿（18%，9/49）是最常见的 2 种组织学类型。12 例行术前选择性动脉栓塞（SAE），24 例行术中主动脉球囊阻断术（ABO）作为术前手术计划。此外，根据肿瘤在骶骨的位置，我们将骶骨良性肿瘤和肿瘤样病变的切除术分为 3 种类型。14 例为Ⅰ型，27 例为Ⅱ型，3 例为Ⅲ型，5 例神经源性肿瘤无法归入该手术分类。10 例患者发生伤口并发症。2 例因 ABO 应用发生股动脉血栓形成，1 例因机械故障发生 ABO 失败。局部复发率为 16%。7 例 GCT 患者和 1 例神经源性肿瘤患者出现局部复发。末次随访时无患者因疾病死亡。对于神经功能评估，神经功能障碍发生率为 12%（6/49）。4 例出现尿失禁，3 例出现大便失禁，3 例出现肠梗阻。进一步行单因素分析显示，术前 SAE 和术中 ABO 对肿瘤和非肿瘤并发症的发生均无显著影响。
提出的手术策略可为骶骨良性肿瘤和骨肿瘤样病变患儿提供良好的治疗效果。术前 SAE 和术中 ABO 可在术中为骶骨高血管良性肿瘤患儿提供保障。
IV 级。
