Bharwani Nandlal, Rathod Priyank, Salunke Abhijeet Ashok, Patel Dhruv, Tripathi Umank, Varun M, Krishana Geeta, Dave Dipesh, Patel Keval, Sharma Mohit, Puj Ketul, Aron Jebin, Bhalerao Raghavendra, Shah Krupa, Deshmukh Suahant, Pandya Shashank
Orthopedic Oncology, Department of Surgical Oncology, The Gujarat Cancer and Research Institute (GCRI), Ahmedabad, India.
Department of Surgical Oncology, The Gujarat Cancer and Research Institute (GCRI), Ahmedabad, India.
Indian J Otolaryngol Head Neck Surg. 2024 Oct;76(5):4270-4279. doi: 10.1007/s12070-024-04835-0. Epub 2024 Jun 29.
Fibrous dysplasia (FD), commonly known as Lichtenstein-Jaffe disease, is a benign fibro-osseous bone disease. Clinical symptoms often include bone pain, deformities, pathological fractures, or nerve compression. Fibrous dysplasia (FD) in the cranio-facial region presents major management concerns because to the risk of deformity, loss of function, and recurrence. The purpose of this study is to demonstrate a single institution's experience managing cranio-facial FD with zolendronic acid, as well as an extensive review of the available literature on the subject. This retrospective study was conducted in the Orthopedic Oncology unit of the Department of Surgical Oncology with a study duration between January 1, 2019, and January 1, 2022. There were seven patients with cranio-facial fibrous dysplasia in the current study. The effects of zolendronic acid were evaluated using clinical assessment, data, radiological findings, biochemical indicators. Furthermore, a comprehensive literature review was conducted in order to compile the current data of cranio-facial FD. The study included seven individuals (five men and two females), four with polyostotic FD and three with cranio-facial FD. The average follow-up duration was 18.75 months. The study found that all parameters improved: the mean VAS score increased from 7 to 1, mean serum calcium levels increased from 8.75 to 8.46 mg/dL, mean serum phosphorus levels increased from 4.46 to 4.17 mg/dL, and serum alkaline phosphatase levels increased from 152 to 93.25 IU/L.A comprehensive literature review was conducted using PubMed and Google Scholar with the following keywords: "Fibrous dysplasia," "Cranio-facial bones," "Bisphosphonate," and "Zolendronic acid." The search included 24 studies published between 2000 and 2022, incorporating all relevant studies available to date. Our study demonstrated the effectiveness of zolendronic acid in the treatment of cranio-facial fibrous dysplasia. Zolendronic acid offers potential as a feasible treatment options in treating cranio-facial FD, with possible advantages including alleviating symptoms, disease progression stabilisation, and morbidity reduction. However, multi-centre prospective randomised study with larger sample numbers and longer follow-up periods are needed in future.
骨纤维异常增殖症(FD),俗称利希滕斯坦 - 贾菲病,是一种良性纤维骨性骨病。临床症状通常包括骨痛、畸形、病理性骨折或神经受压。颅面部的骨纤维异常增殖症(FD)由于存在畸形、功能丧失和复发的风险,带来了主要的治疗难题。本研究的目的是展示一家机构使用唑来膦酸治疗颅面部FD的经验,并对该主题的现有文献进行广泛综述。这项回顾性研究在外科肿瘤学系的骨肿瘤病房进行,研究时间段为2019年1月1日至2022年1月1日。本研究中有7例颅面部骨纤维异常增殖症患者。使用临床评估、数据、放射学检查结果、生化指标评估唑来膦酸的疗效。此外,进行了全面的文献综述以汇编颅面部FD的当前数据。该研究纳入了7名个体(5名男性和2名女性),4例为多骨型FD,3例为颅面部FD。平均随访时间为18.75个月。研究发现所有参数均有所改善:视觉模拟评分(VAS)均值从7分降至1分,血清钙均值从8.75毫克/分升升至8.46毫克/分升,血清磷均值从4.46毫克/分升升至4.17毫克/分升,血清碱性磷酸酶水平从152国际单位/升降至93.25国际单位/升。使用PubMed和谷歌学术搜索引擎,以“骨纤维异常增殖症”“颅面骨”“双膦酸盐”和“唑来膦酸”为关键词进行了全面的文献综述。检索纳入了2000年至2022年发表的24项研究,涵盖了迄今为止所有相关研究。我们的研究证明了唑来膦酸在治疗颅面部骨纤维异常增殖症方面的有效性。唑来膦酸作为治疗颅面部FD的一种可行治疗选择具有潜力,可能的优势包括缓解症状、稳定疾病进展和降低发病率。然而,未来需要进行更大样本量和更长随访期的多中心前瞻性随机研究。