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儿童恶性肿瘤放疗后发生的骨软骨瘤:针对骨骼副作用加强咨询的作用

Osteochondromas after radiation for pediatric malignancies: a role for expanded counseling for skeletal side effects.

作者信息

King Elizabeth A, Hanauer David A, Choi Sung Won, Jong Nahbee, Hamstra Daniel A, Li Ying, Farley Frances A, Caird Michelle S

机构信息

Departments of *Orthopaedic Surgery †Pediatrics & Communicable Diseases ‡Radiation Oncology, University of Michigan, Mott Children's Hospital, Ann Arbor, MI.

出版信息

J Pediatr Orthop. 2014 Apr-May;34(3):331-5. doi: 10.1097/BPO.0000000000000081.

Abstract

BACKGROUND

A relationship has been reported between total body irradiation (TBI) and later development of osteochondromas in children who receive radiation therapy as conditioning before hematopoietic stem cell transplantation (HSCT). The goal of this study was to better characterize osteochondromas occurring in these children.

METHODS

We identified all children (0 to 18 y) who received an allogeneic HSCT and TBI from 2000 to 2012 from a blood and marrow transplant (BMT) database. Thereafter, we identified those who developed osteochondromas through a chart review. In addition, we searched for diagnosis and operative codes from 1996 to 2012 in our pediatric orthopaedic clinical records, isolating osteochondroma patients with a history of radiation exposure.

RESULTS

Four patients who underwent allogeneic HSCT and were later diagnosed with osteochondromas were identified from the BMT database (N=233 children); all 4 were among a group of 72 patients who received TBI. Three patients were identified from orthopaedic records. The cohort included 5 boys and 2 girls with acute lymphoblastic leukemia (N=5) or neuroblastoma (N=2), diagnosed at a median age of 2.0 years. Therapy for all patients included chemotherapy, radiation therapy (TBI, N=5; abdominal, N=2), and HSCT. A diagnosis of osteochondroma was made at a median age of 11.7 years (range, 5 to 16 y), on average 8.6 years after radiation therapy. Diagnosis was incidental in 2 patients and secondary to symptoms (pain or genu valgum) in 5. Locations of osteochondromas were the proximal tibia (N=3), distal tibia, distal femur, distal ulna, and the distal phalanx (N=1 each). Three patients underwent surgical resection.

CONCLUSIONS

Children may be more likely to develop osteochondromas after early exposure to radiation therapy, which may cause pain and require surgical resection. To the best of our knowledge, this is the first reported case of a radiation-induced osteochondroma causing lower extremity malalignment. Patients typically present to the pediatric orthopaedist's attention when symptomatic, but there may be an expanded role for counseling for potential for long-term skeletal effects in this group.

LEVEL OF EVIDENCE

Level IV, case series.

摘要

背景

据报道,在接受造血干细胞移植(HSCT)前作为预处理进行全身照射(TBI)的儿童中,TBI与后来骨软骨瘤的发生之间存在关联。本研究的目的是更好地描述这些儿童中发生的骨软骨瘤。

方法

我们从血液和骨髓移植(BMT)数据库中识别出2000年至2012年期间接受异基因HSCT和TBI的所有儿童(0至18岁)。此后,我们通过病历审查确定了那些发生骨软骨瘤的儿童。此外,我们在儿科骨科临床记录中搜索了1996年至2012年的诊断和手术编码,筛选出有辐射暴露史的骨软骨瘤患者。

结果

从BMT数据库(N = 233名儿童)中识别出4名接受异基因HSCT且后来被诊断为骨软骨瘤的患者;这4名患者均在接受TBI的72名患者中。从骨科记录中识别出3名患者。该队列包括5名男孩和2名女孩,患有急性淋巴细胞白血病(N = 5)或神经母细胞瘤(N = 2),诊断时的中位年龄为2.0岁。所有患者的治疗包括化疗、放疗(TBI,N = 5;腹部放疗,N = 2)和HSCT。骨软骨瘤的诊断中位年龄为11.7岁(范围5至16岁),平均在放疗后8.6年。2例患者为偶然诊断,5例因症状(疼痛或膝外翻)继发诊断。骨软骨瘤的部位为胫骨近端(N = 3)、胫骨远端、股骨远端、尺骨远端和远端指骨(各N = 1)。3例患者接受了手术切除。

结论

儿童在早期接受放射治疗后可能更易发生骨软骨瘤,这可能导致疼痛并需要手术切除。据我们所知,这是首例报道的因辐射诱发骨软骨瘤导致下肢畸形的病例。患者通常在出现症状时引起儿科骨科医生的注意,但对于该群体潜在的长期骨骼影响,咨询可能具有更广泛的作用。

证据水平

IV级,病例系列。

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