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戈勒姆-斯托特病：采用未获批准的抑制-缓解疗法进行多棒腰椎重建术。

Gorham-Stout disease: A multirod lumbar reconstruction with off-label suppression-remission therapy.

作者信息

Krishnan Ajay, Raj Aditya, Degulmadi Devanand, Mayi Shivanand, Rai Raviranjan, Bali Shiv Kumar, Parmar Vatsal, Amin Prarthan Chirag, Krishnan Preety, Dave Mirant, Dave Bharat

机构信息

Department of Spine Surgery, Stavya Spine Hospital and Research Institute, Ahmedabad, Gujarat, India.

出版信息

Surg Neurol Int. 2022 Apr 8;13:136. doi: 10.25259/SNI_221_2022. eCollection 2022.

DOI:10.25259/SNI_221_2022
PMID:35509593
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9062908/
Abstract

BACKGROUND

Gorham-Stout disease (GSD), a fibro-lymphovascular entity in which tissue replaces the bone leading to massive osteolysis and its sequelae, rarely leads to spinal deformity/instability and neurological deficits. Here, we report a 12-year-old female who was diagnosed and treated for GSD.

CASE DESCRIPTION

A 12-year-old female presented with back pain, and the inability to walk, sit, or stand attributed to three MR/CT documented L2-L4 lumbar vertebral collapses. Closed biopsies were negative. However, an open biopsy diagnosed GSD. She underwent a dorsal-lumbar-to-pelvis fusion (i.e., T5-T12 through L5/S1/S2) using multilevel pedicle screw/rod stabilization and human leukocyte antigens (HLAs) matched allograft (i.e. from her father). Postoperatively, she was treated with "off-label" teriparatide injections, bisphosphonates, and sirolimus. Four years later, while continuing the bisphosphonate therapy, she remained stable.

CONCLUSION

Surgical multirod stabilization from T5 to S2, supplemented with HLA compatible allograft, and multiple medical "off-label" therapies (i.e., teriparatide, sirolimus, and bisphosphonates) led to a good 4-year outcome in a 12-year-old female with GSD.

摘要

背景

戈勒姆-斯托特病（GSD）是一种纤维-淋巴血管性疾病，其中组织替代骨骼导致大量骨质溶解及其后遗症，很少导致脊柱畸形/不稳定和神经功能缺损。在此，我们报告一名12岁女性，她被诊断患有GSD并接受了治疗。

病例描述

一名12岁女性因背部疼痛就诊，因磁共振成像/计算机断层扫描（MR/CT）记录的L2-L4腰椎椎体塌陷而无法行走、坐立或站立。闭合活检结果为阴性。然而，开放活检诊断为GSD。她接受了胸腰段至骨盆融合术（即T5-T12至L5/S1/S2），采用多级椎弓根螺钉/棒稳定术，并使用与人类白细胞抗原（HLA）匹配的同种异体移植物（即来自她父亲的移植物）。术后，她接受了“超说明书用药”的特立帕肽注射、双膦酸盐和西罗莫司治疗。四年后，在继续双膦酸盐治疗的情况下，她病情保持稳定。

结论

对于一名患有GSD的12岁女性，从T5到S2的手术多棒稳定术，辅以HLA相容的同种异体移植物，以及多种“超说明书用药”治疗（即特立帕肽、西罗莫司和双膦酸盐），带来了良好的4年治疗效果。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c236/9062908/bbe71ed40527/SNI-13-136-g012.jpg
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