Dhillon Charanjit Singh, Tantry Raviraj, Ega Shrikant Rajeshwari, Pophale Chetan, Medagam Narendra Reddy, Chhasatia Nilay
Department of Spine services, MIOT International, Chennai. Tamil Nadu, India.
J Orthop Case Rep. 2020 Dec;10(9):28-32. doi: 10.13107/jocr.2020.v10.i09.1892.
Langerhans cell histiocytosis (LCH) in spine is a benign disorder that mainly affects children and is rare in adults. The treatment of LCH in adults is still controversial. The literature is drought with reports regarding management of LCH in adults with pathological fracture. We report a case of LCH at L5 vertebra in an adult patient treated with posterior stabilization, decompression, and anterior corpectomy and reconstruction.
A 30-year-old manual laborer working in Middle East, presented to us with severe pain in the lower back (VAS-8) with the right lower limb radiculopathy for 6 months. Radiological investigations revealed to have a solitary osteolytic lesion with pathological fracture at L5 vertebral body. MRI showed hyperintense lesion in T2 sagittal images and hypointense in T1 sagittal images in L5 vertebral body. PET scan showed metabolically active lesion involving L5 vertebra body and right ischium. CT-guided biopsy from L5 vertebral body was performed, but was inconclusive. The patient underwent surgical management in the form of posterior stabilization L4-S1 and transpedicular biopsy. The sample was sent for frozen section and confirmed the presence of neoplasia but did not provide sufficient information about the nature of pathology. Intraoperatively, the decision was made to do anterior excision biopsy, corpectomy, and reconstruction with titanium mesh cage filled with cement. The precise diagnosis of LCH was established on histopathological examination and confirmed with immunohistochemistry positivity for CD1a and S100. The patient had immediate relief of his back pain and radicular pain. He was able to resume his daily activities at 1 month after the surgery. At 2-year follow-up patient was asymptomatic and no local recurrence was noticed.
Surgical excisionfor LCH in adults should be considered in patients with refractory low back pain with pathological fracture, neurological deficits, or spinal instability.
脊柱朗格汉斯细胞组织细胞增多症(LCH)是一种主要影响儿童的良性疾病,在成人中较为罕见。成人LCH的治疗仍存在争议。关于成人LCH合并病理性骨折的治疗报道较少。我们报告一例成人患者L5椎体LCH,采用后路稳定、减压、前路椎体切除及重建治疗。
一名30岁在中东工作的体力劳动者,因下背部剧痛(视觉模拟评分8分)伴右下肢神经根病6个月前来就诊。影像学检查显示L5椎体有一个孤立性溶骨性病变并伴有病理性骨折。MRI显示L5椎体在T2矢状位图像上为高信号病变,在T1矢状位图像上为低信号。PET扫描显示L5椎体和右坐骨有代谢活跃病变。对L5椎体进行了CT引导下活检,但结果不明确。患者接受了L4-S1后路稳定和经椎弓根活检的手术治疗。样本送去做冰冻切片检查,证实存在肿瘤,但未提供足够的病理性质信息。术中决定进行前路切除活检、椎体切除,并用填充骨水泥的钛网笼进行重建。经组织病理学检查确诊为LCH,并经CDla和S100免疫组化阳性证实。患者的背痛和神经根痛立即缓解。术后1个月他能够恢复日常活动。在2年的随访中,患者无症状,未发现局部复发。
对于患有顽固性下背痛合并病理性骨折、神经功能缺损或脊柱不稳定的成人LCH患者,应考虑手术切除。