BACKGROUND For patients with acute paraplegia caused by spinal giant cell tumor(GCT)who require emergency decompressive surgery,there is still a lack of relevant reports on surgical options.This study is the first to ...BACKGROUND For patients with acute paraplegia caused by spinal giant cell tumor(GCT)who require emergency decompressive surgery,there is still a lack of relevant reports on surgical options.This study is the first to present the case of an acute paraplegic patient with a thoracic spinal GCT who underwent an emergency total en bloc spondylectomy(TES).Despite tumor recurrence,three-level TES was repeated after denosumab therapy.CASE SUMMARY A 27-year-old female patient who underwent single-level TES in an emergency presented with sudden severe back pain and acute paraplegia due to a thoracic spinal tumor.After emergency TES,the patient's spinal cord function recovered,and permanent paralysis was avoided.The postoperative histopathological examination revealed that the excised neoplasm was a rare GCT.Unfortunately,the tumor recurred 9 months after the first surgery.After 12 months of denosumab therapy,the tumor size was reduced,and tumor calcification.To prevent recurrent tumor progression and provide a possible cure,a three-level TES was performed again.The patient returned to an active lifestyle 1 month after the second surgery,and no recurrence of GCT was found at the last follow-up.CONCLUSION This patient with acute paraplegia underwent TES twice,including once in an emergency,and achieved good therapeutic results.TES in emergency surgery is feasible and safe when conditions permit;however,it may increase the risk of tumor recurrence.展开更多
Background: Giant cell tumor (GCT) is a common benign tumor of the appendicular and axial skeleton that represents 5% of all primary bone tumors. In recent years, the combination of conventional aggressive curettage w...Background: Giant cell tumor (GCT) is a common benign tumor of the appendicular and axial skeleton that represents 5% of all primary bone tumors. In recent years, the combination of conventional aggressive curettage with targeted adjuvant anti-osteoclastic agents including bisphosphonates and denosumab have led to lower recurrence rates in patients with GCT in a small number of retrospective case series. Furthermore, efficacy of the same anti-osteoclastic agents has been shown in cases of unresectable GCT of bone, leading to decreased rates of tumor progression and stabilization of disease. This review assesses whether the current literature weakly, moderately, or strongly supports a targeted systemic treatment as the standard of care in patients with GCT. Methods: We conducted a current search of the MEDLINE database for literature pertaining to systemic GCT treatment. Our inclusion criteria were as follows: 1) studies that reported on a series of patients with resectable or unresectable cases of GCT;2) a subset of patients must have been treated with systemic bisphosphonate or RANK-L inhibitor therapy;3) each series had a minimum of 10 patients with histopathologically confirmed GCT;4) each series stated their follow-up period. Results: Overall 6 studies, reporting on a total of 487 patients, were selected for inclusion in this review. For analysis, these 6 retrospective studies were subdivided into series where all GCT patients had resectable tumors (n = 4) and series where patients had a mix of resectable and unresectable tumors (n = 2). The overall recurrence rate of GCT in patients with resectable tumors treated with adjuvant systemic bisphosphonates was 6.7% compared to 48.4% in patients not treated with adjuvant systemic bisphosphonates (p 0.0001). In patients with both resectable and unresectable primary aggressive, recurrent, or metastatic GCT disease, systemic bisphosphonate and denosumab demonstrated good efficacy with decreased rates of disease progression and recurrence. In general the side effects of bisphosphonates were mild while denosumab had a more severe side effect profile. Conclusions: Systemic treatment with bisphosphonates or denosumab in cases of GCT is promising, but there is a lack of high-level evidence with sufficient follow-up supporting their use. We believe the current literature provides moderate support to recommend a short course of adjuvant peri-operative systemic bisphosphonate treatment for patients with resectable primary GCT and moderate support to recommend adjuvant peri-operative (resectable) and non-operative (unresectable) use of denosumab in cases of primary aggressive, recurrent, or metastatic GCT. With either systemic treatment, patients should be well counseled on all potential side effects in addition to alternative treatment, which includes the option of no systemic treatment.展开更多
Background: Giant cell tumors of the lumbar spine are rare and complete resection without major functional comprise is challenging despite advancements in spine surgery techniques. Radiation therapy has been an option...Background: Giant cell tumors of the lumbar spine are rare and complete resection without major functional comprise is challenging despite advancements in spine surgery techniques. Radiation therapy has been an option in such cases;however there are high concerns for associated high small bowel toxicity and lack of dose escalation to achieve local control. With advent of intensity modulated radiation therapy (IMRT) it is now possible to give high radiation dose to tumor with minimal toxicity. Herein we present a rare case of giant cell tumor of fourth lumbar (L4) vertebra treated with RapidArc intensity modulated arc therapy (IMAT) with relevant review of literature. Methods: A 30-year-old female had low back pain for 6 months. She underwent surgical decompression at another hospital as having a L4-5 protruded intervertebral disc 2 months previously, but her back pain progressed with weakness of both legs with restricted movement. Radiological and pathological work-up confirmed the diagnosis of giant cell tumor of L4 vertebra. She refused further surgery and was referred to us for radiotherapy. Treatment plans for prescribed radiation dose of 59.4 Gy in 30 fractions were made by 3DCRT and RapidArc IMAT and comparison was made. Student’s unpaired t test was used to determine the significance of the difference between two plans in terms of dose to the tumor and small bowel. A p value of 0.05 was considered statistically significant. Related literature was searched. Results: In RapidArc IMAT and 3DCRT plans mean doses to planning treatment volume (PTV) were 61.24 Gy (55.98 - 66.23) and 60.71 Gy (49.87 - 63.74) respectively (p 0.04) and mean doses to small bowel were found lesser in RapidArc plan [14.78 Gy (range: 0.39 - 53.15)] as compared to 3DCRT plan. Patient was started on RapidArc IMRT and she completed the course without any major sequelae. Conclusion: Lumbar spine giant cell tumors are rare and complete resection is often not possible. RapidArc IMAT is a feasible option for such patients to deliver high dose radiation to achieve good local control with marked symptom relief and without severe toxicity.展开更多
Primary giant-cell tumors rarely arise in the common bile duct.We herein report a case of primary giantcell tumor of the common bile duct.The patient was an 81-year-old male who was diagnosed with a welldefined 1.2-cm...Primary giant-cell tumors rarely arise in the common bile duct.We herein report a case of primary giantcell tumor of the common bile duct.The patient was an 81-year-old male who was diagnosed with a welldefined 1.2-cm mass projecting into the lumen of the middle common bile duct.Excision of the gallbladder and extrahepatic bile duct and a Roux-en-Y cholangiojejunostomy were performed.Histologically,the tumor had no association with carcinomas of epithelial origin and was similar to giant-cell tumors of the bone.The tumor consisted of a mixture of mononuclear and multinucleated osteoclast-like giant cells.The mononuclear cells showed no atypical features,and their nuclei were similar to those of the multinucleated giant cells.CD68 was expressed on the mononuclear and multinucleated osteoclast-like giant cells,whereas CD163 immunoreactivity was restricted to the mononuclear cells.Six months after the operation,the patient was still alive and had no recurrence.The interest of this case lies in the rarity of this entity,the difficulty of preoperative diagnosis,and this tumor's possible confusion with other malignant tumors.展开更多
<p style="text-align:justify;"> <span style="font-family:Verdana;">Giant cell tumor of the wrist is a rare, benign and usually symptomatic condition. The discovery is sometimes made fol...<p style="text-align:justify;"> <span style="font-family:Verdana;">Giant cell tumor of the wrist is a rare, benign and usually symptomatic condition. The discovery is sometimes made following a medical imaging examination or a painful symptomatology or more often a visible or palpable swelling with or without vascular and/or nerve compression. At an advanced stage, the X-ray is of paramount importance. The well codified complete surgical resection is part of the therapeutic arsenal.</span><span style="font-family:Verdana;"> </span><span style="font-family:Verdana;">We present a clinical case report of a young woman with a giant cell tumor localized in the wrist in N’Djamena, Chad. This case concerns a 25-year-old patient who presented in July 2020 of a painful swelling lateral to her left wrist bone and whose X-ray radiography showed lysis of the cortical bone in the lower third of the ulna. After the operative resection of the tumor mass, the pathological examination of the operative specimen revealed the diagnosis of a giant cell tumor.</span><span style="font-family:Verdana;"> </span><span style="font-family:Verdana;">A giant cell tumor is a benign condition, with a few symptoms and the location at the ulna is exceptional. Complete surgical resection is a viable treatment option.</span> </p>展开更多
目的回顾性分析股骨近端骨巨细胞瘤患者的手术方式,验证GTOC临床评分系统的有效性。方法2011年1月至2022年12月,共收集浙江大学医学院附属第二医院股骨近端骨巨细胞瘤患者45例,根据中国骨巨细胞瘤协作组成成员(Member of the Chinese As...目的回顾性分析股骨近端骨巨细胞瘤患者的手术方式,验证GTOC临床评分系统的有效性。方法2011年1月至2022年12月,共收集浙江大学医学院附属第二医院股骨近端骨巨细胞瘤患者45例,根据中国骨巨细胞瘤协作组成成员(Member of the Chinese Association for Giant Cell tumor of Bone,GTOC)临床评分系统进行评分,统计分析GTOC临床评分系统与手术方式、术后并发症、下肢功能的关系,采用MSTS93下肢功能评分系统评估患肢术后功能,分析影响股骨近端骨巨细胞瘤手术方式选择的因素,对扩大刮除术与大段切除术进行对比分析,从而验证GTOC临床评分系统的有效性。结果45例患者随访时间12~86个月,平均25.7个月。共出现13例并发症,总的发生率28.9%,复发2例,总复发率为4.4%。单因素及多因素Cox回归分析显示影响手术方式选择的因素是病理性骨折和病变部位(P<0.05)。统计资料分析显示,高分组与低分组的手术方式差异有统计学意义(χ^(2)=8.557,P<0.05),呈正相关关系(Pearson相关系数R=0.436,P=0.003)。对扩大刮除术与瘤段切除重建术进行对比分析,两种手术方式的术后MSTS功能评分、手术时间有明显差异(P<0.05)。结论GTOC临床评分系统涵盖了手术方式选择的主要影响因素,对于股骨近端骨巨细胞瘤患者在临床治疗手术方案的选择中具有指导价值。展开更多
基金Supported by The Shanghai Municipal Health Commission Clinical Research Project,No.202140140.
文摘BACKGROUND For patients with acute paraplegia caused by spinal giant cell tumor(GCT)who require emergency decompressive surgery,there is still a lack of relevant reports on surgical options.This study is the first to present the case of an acute paraplegic patient with a thoracic spinal GCT who underwent an emergency total en bloc spondylectomy(TES).Despite tumor recurrence,three-level TES was repeated after denosumab therapy.CASE SUMMARY A 27-year-old female patient who underwent single-level TES in an emergency presented with sudden severe back pain and acute paraplegia due to a thoracic spinal tumor.After emergency TES,the patient's spinal cord function recovered,and permanent paralysis was avoided.The postoperative histopathological examination revealed that the excised neoplasm was a rare GCT.Unfortunately,the tumor recurred 9 months after the first surgery.After 12 months of denosumab therapy,the tumor size was reduced,and tumor calcification.To prevent recurrent tumor progression and provide a possible cure,a three-level TES was performed again.The patient returned to an active lifestyle 1 month after the second surgery,and no recurrence of GCT was found at the last follow-up.CONCLUSION This patient with acute paraplegia underwent TES twice,including once in an emergency,and achieved good therapeutic results.TES in emergency surgery is feasible and safe when conditions permit;however,it may increase the risk of tumor recurrence.
文摘Background: Giant cell tumor (GCT) is a common benign tumor of the appendicular and axial skeleton that represents 5% of all primary bone tumors. In recent years, the combination of conventional aggressive curettage with targeted adjuvant anti-osteoclastic agents including bisphosphonates and denosumab have led to lower recurrence rates in patients with GCT in a small number of retrospective case series. Furthermore, efficacy of the same anti-osteoclastic agents has been shown in cases of unresectable GCT of bone, leading to decreased rates of tumor progression and stabilization of disease. This review assesses whether the current literature weakly, moderately, or strongly supports a targeted systemic treatment as the standard of care in patients with GCT. Methods: We conducted a current search of the MEDLINE database for literature pertaining to systemic GCT treatment. Our inclusion criteria were as follows: 1) studies that reported on a series of patients with resectable or unresectable cases of GCT;2) a subset of patients must have been treated with systemic bisphosphonate or RANK-L inhibitor therapy;3) each series had a minimum of 10 patients with histopathologically confirmed GCT;4) each series stated their follow-up period. Results: Overall 6 studies, reporting on a total of 487 patients, were selected for inclusion in this review. For analysis, these 6 retrospective studies were subdivided into series where all GCT patients had resectable tumors (n = 4) and series where patients had a mix of resectable and unresectable tumors (n = 2). The overall recurrence rate of GCT in patients with resectable tumors treated with adjuvant systemic bisphosphonates was 6.7% compared to 48.4% in patients not treated with adjuvant systemic bisphosphonates (p 0.0001). In patients with both resectable and unresectable primary aggressive, recurrent, or metastatic GCT disease, systemic bisphosphonate and denosumab demonstrated good efficacy with decreased rates of disease progression and recurrence. In general the side effects of bisphosphonates were mild while denosumab had a more severe side effect profile. Conclusions: Systemic treatment with bisphosphonates or denosumab in cases of GCT is promising, but there is a lack of high-level evidence with sufficient follow-up supporting their use. We believe the current literature provides moderate support to recommend a short course of adjuvant peri-operative systemic bisphosphonate treatment for patients with resectable primary GCT and moderate support to recommend adjuvant peri-operative (resectable) and non-operative (unresectable) use of denosumab in cases of primary aggressive, recurrent, or metastatic GCT. With either systemic treatment, patients should be well counseled on all potential side effects in addition to alternative treatment, which includes the option of no systemic treatment.
文摘Background: Giant cell tumors of the lumbar spine are rare and complete resection without major functional comprise is challenging despite advancements in spine surgery techniques. Radiation therapy has been an option in such cases;however there are high concerns for associated high small bowel toxicity and lack of dose escalation to achieve local control. With advent of intensity modulated radiation therapy (IMRT) it is now possible to give high radiation dose to tumor with minimal toxicity. Herein we present a rare case of giant cell tumor of fourth lumbar (L4) vertebra treated with RapidArc intensity modulated arc therapy (IMAT) with relevant review of literature. Methods: A 30-year-old female had low back pain for 6 months. She underwent surgical decompression at another hospital as having a L4-5 protruded intervertebral disc 2 months previously, but her back pain progressed with weakness of both legs with restricted movement. Radiological and pathological work-up confirmed the diagnosis of giant cell tumor of L4 vertebra. She refused further surgery and was referred to us for radiotherapy. Treatment plans for prescribed radiation dose of 59.4 Gy in 30 fractions were made by 3DCRT and RapidArc IMAT and comparison was made. Student’s unpaired t test was used to determine the significance of the difference between two plans in terms of dose to the tumor and small bowel. A p value of 0.05 was considered statistically significant. Related literature was searched. Results: In RapidArc IMAT and 3DCRT plans mean doses to planning treatment volume (PTV) were 61.24 Gy (55.98 - 66.23) and 60.71 Gy (49.87 - 63.74) respectively (p 0.04) and mean doses to small bowel were found lesser in RapidArc plan [14.78 Gy (range: 0.39 - 53.15)] as compared to 3DCRT plan. Patient was started on RapidArc IMRT and she completed the course without any major sequelae. Conclusion: Lumbar spine giant cell tumors are rare and complete resection is often not possible. RapidArc IMAT is a feasible option for such patients to deliver high dose radiation to achieve good local control with marked symptom relief and without severe toxicity.
文摘Primary giant-cell tumors rarely arise in the common bile duct.We herein report a case of primary giantcell tumor of the common bile duct.The patient was an 81-year-old male who was diagnosed with a welldefined 1.2-cm mass projecting into the lumen of the middle common bile duct.Excision of the gallbladder and extrahepatic bile duct and a Roux-en-Y cholangiojejunostomy were performed.Histologically,the tumor had no association with carcinomas of epithelial origin and was similar to giant-cell tumors of the bone.The tumor consisted of a mixture of mononuclear and multinucleated osteoclast-like giant cells.The mononuclear cells showed no atypical features,and their nuclei were similar to those of the multinucleated giant cells.CD68 was expressed on the mononuclear and multinucleated osteoclast-like giant cells,whereas CD163 immunoreactivity was restricted to the mononuclear cells.Six months after the operation,the patient was still alive and had no recurrence.The interest of this case lies in the rarity of this entity,the difficulty of preoperative diagnosis,and this tumor's possible confusion with other malignant tumors.
文摘<p style="text-align:justify;"> <span style="font-family:Verdana;">Giant cell tumor of the wrist is a rare, benign and usually symptomatic condition. The discovery is sometimes made following a medical imaging examination or a painful symptomatology or more often a visible or palpable swelling with or without vascular and/or nerve compression. At an advanced stage, the X-ray is of paramount importance. The well codified complete surgical resection is part of the therapeutic arsenal.</span><span style="font-family:Verdana;"> </span><span style="font-family:Verdana;">We present a clinical case report of a young woman with a giant cell tumor localized in the wrist in N’Djamena, Chad. This case concerns a 25-year-old patient who presented in July 2020 of a painful swelling lateral to her left wrist bone and whose X-ray radiography showed lysis of the cortical bone in the lower third of the ulna. After the operative resection of the tumor mass, the pathological examination of the operative specimen revealed the diagnosis of a giant cell tumor.</span><span style="font-family:Verdana;"> </span><span style="font-family:Verdana;">A giant cell tumor is a benign condition, with a few symptoms and the location at the ulna is exceptional. Complete surgical resection is a viable treatment option.</span> </p>
文摘目的回顾性分析股骨近端骨巨细胞瘤患者的手术方式,验证GTOC临床评分系统的有效性。方法2011年1月至2022年12月,共收集浙江大学医学院附属第二医院股骨近端骨巨细胞瘤患者45例,根据中国骨巨细胞瘤协作组成成员(Member of the Chinese Association for Giant Cell tumor of Bone,GTOC)临床评分系统进行评分,统计分析GTOC临床评分系统与手术方式、术后并发症、下肢功能的关系,采用MSTS93下肢功能评分系统评估患肢术后功能,分析影响股骨近端骨巨细胞瘤手术方式选择的因素,对扩大刮除术与大段切除术进行对比分析,从而验证GTOC临床评分系统的有效性。结果45例患者随访时间12~86个月,平均25.7个月。共出现13例并发症,总的发生率28.9%,复发2例,总复发率为4.4%。单因素及多因素Cox回归分析显示影响手术方式选择的因素是病理性骨折和病变部位(P<0.05)。统计资料分析显示,高分组与低分组的手术方式差异有统计学意义(χ^(2)=8.557,P<0.05),呈正相关关系(Pearson相关系数R=0.436,P=0.003)。对扩大刮除术与瘤段切除重建术进行对比分析,两种手术方式的术后MSTS功能评分、手术时间有明显差异(P<0.05)。结论GTOC临床评分系统涵盖了手术方式选择的主要影响因素,对于股骨近端骨巨细胞瘤患者在临床治疗手术方案的选择中具有指导价值。
