Camurati–Engelmann disease (CED) is a rare, progressive diaphyseal dysplasia characterized as diaphyseal hyperostosis and sclerosis of the long bones. Corticosteroids, bisphosphonates, and losartan have been reported to be effective systemic medications used to reduce CED symptoms. There are no reports of osteoblastoma in patients with CED, and osteoblastoma in the distal radius is rare. We present a patient diagnosed with CED, based on radiological and histological examinations, at 11 years old. At 22 years old, she experienced severe pain in her right forearm and was treated with bisphosphonate, losartan, and prednisolone; however, the pain continued. An expansive and sclerotic lesion at the distal radius was observed on radiography. A follow-up plain radiograph indicated that the lesion was growing. Fluorodeoxyglucose positron emission tomography revealed solitary, intense radiotracer uptake, and a biopsy and surgical resection were performed due to suspected malignancy. Pathologic analysis showed anastomosing bony trabeculae rimmed by osteoblasts observed in a loose fibrovascular stroma. The lesion was diagnosed as an osteoblastoma. Following bone excision and artificial bone grafting, the patient’s severe pain almost completely disappeared. At final follow-up, no evidence of osteoblastoma recurrence was noted. To our knowledge, this is the first case report of osteoblastoma arising in a patient with CED. Bone excision and artificial bone grafting may be a treatment option for local symptomatic osteoblastoma in patients with CED.

Camurati-Engelmann 病 (CED) 是一种罕见的进行性骨干发育不良，其特征是骨干骨质增生和长骨硬化。据报道，皮质类固醇、双膦酸盐和氯沙坦是用于减轻 CED 症状的有效全身药物。CED患者尚无成骨细胞瘤的报道，桡骨远端成骨细胞瘤少见。我们介绍了一名 11 岁时根据放射学和组织学检查诊断为 CED 的患者。22 岁时，她的右前臂出现剧烈疼痛，接受双膦酸盐、氯沙坦和泼尼松龙治疗；然而，痛苦仍在继续。放射线照相术观察到桡骨远端有一个扩大和硬化的病变。后续平片显示病变正在增长。氟脱氧葡萄糖正电子发射断层扫描显示孤立的、强烈的放射性示踪剂摄取，由于疑似恶性肿瘤而进行了活检和手术切除。病理学分析显示，在松散的纤维血管基质中观察到由成骨细胞包围的吻合骨小梁。病变被诊断为骨母细胞瘤。进行骨切除和人工骨移植后，患者的剧烈疼痛几乎完全消失。在最后的随访中，没有发现成骨细胞瘤复发的证据。据我们所知，这是首例 CED 患者发生骨母细胞瘤的病例报告。骨切除和人工骨移植可能是 CED 患者局部症状性成骨细胞瘤的治疗选择。由于疑似恶性肿瘤，进行了活检和手术切除。病理学分析显示，在松散的纤维血管基质中观察到由成骨细胞包围的吻合骨小梁。病变被诊断为骨母细胞瘤。进行骨切除和人工骨移植后，患者的剧烈疼痛几乎完全消失。在最后的随访中，没有发现成骨细胞瘤复发的证据。据我们所知，这是首例 CED 患者发生骨母细胞瘤的病例报告。骨切除和人工骨移植可能是 CED 患者局部症状性成骨细胞瘤的治疗选择。由于疑似恶性肿瘤，进行了活检和手术切除。病理学分析显示，在松散的纤维血管基质中观察到由成骨细胞包围的吻合骨小梁。病变被诊断为骨母细胞瘤。进行骨切除和人工骨移植后，患者的剧烈疼痛几乎完全消失。在最后的随访中，没有发现成骨细胞瘤复发的证据。据我们所知，这是首例 CED 患者发生骨母细胞瘤的病例报告。骨切除和人工骨移植可能是 CED 患者局部症状性成骨细胞瘤的治疗选择。进行骨切除和人工骨移植后，患者的剧烈疼痛几乎完全消失。在最后的随访中，没有发现成骨细胞瘤复发的证据。据我们所知，这是首例 CED 患者发生骨母细胞瘤的病例报告。骨切除和人工骨移植可能是 CED 患者局部症状性成骨细胞瘤的治疗选择。进行骨切除和人工骨移植后，患者的剧烈疼痛几乎完全消失。在最后的随访中，没有发现成骨细胞瘤复发的证据。据我们所知，这是首例 CED 患者发生骨母细胞瘤的病例报告。骨切除和人工骨移植可能是 CED 患者局部症状性成骨细胞瘤的治疗选择。