A lung metastasis from giant cell tumor of bone at eight years after primary resection.

We report a case of extensive pulmonary metastasis from a histologically benign giant cell tumor. A 34-year-old woman had undergone tumor resection, curettage and artificial bone grafting for giant cell tumor in the left tibia. At eight years after surgery, a chest radiograph revealed an extensive tumor shadow in the right thoracic cavity. Tumor resection with right upper and middle bilobectomy was performed. Its histological features were consistent with those of the primary tumor of the tibia. Distant metastases from a giant cell tumor of bone are rare, with only 50 reported cases. Metastasis has occurred mainly within 3 years after the primary resection. However, in 20% of reported cases, metastasis occurred after 5 years or longer. Long-term follow-up and careful observation for distant metastasis are necessary for this histologically benign disease.     

带蒂腓骨移位修复胫骨股骨长段骨缺损

目的 总结修复胫骨上段,中段及股骨下段长段骨缺损的效果。方法 自１９７４年５月￣１９９７年８月应用四种带蒂腓骨局部移位术修复胫骨,股骨头段骨缺损２５例,其中胫骨上端良性骨肿瘤,瘤段切除后带蒂腓骨移位,膝关节融合９例,胫骨干良性长段骨肿瘤或瘤样病变者,截除后带肌蒂腓骨移位,行胫腓骨融合术９例,胫骨中下段广泛良性骨肿瘤或瘤样病变,截除后血管蒂腓骨向远端内侧移位,胫腓骨融合２例,股骨下端良性骨肿瘤,切除     

Kotz modular femur and tibia replacement: 28 tumor cases followed for 3 (1-8) years

In the period 1984-1991, 33 lower limb reconstructions were performed with an uncemented Kotz modular femur tibia reconstruction (KMFTR) prosthesis after resection of 32 malignant bone tumors and 1 benign giant cell tumor. Tumor localization was proximal femur in 12, distal femur in 17, and proximal tibia in 4. The mean age of the patients was 38 years. 28 patients with a minimum follow-up of 1 year were studied after 3 (1-8) years. 7 patients were reoperated because of complications. Good or excellent results were obtained in 6 of the 10 proximal and 13 of the 14 distal femur reconstructions. In 2 of the 4 patients undergoing combined distal femur and proximal tibia resection, a deep infection developed, and above-knee amputation was performed. One local recurrence occurred after proximal femur resection.     

酒精灭活瘤段骨复合假体治疗膝关节周围骨巨细胞瘤的中期随访研究

 目的 探计膝关节周围骨巨细胞瘤整块切除,酒精灭活瘤段骨复合假体重建的手术技术及预后。方法 2007年1月至2008年10月,对8例膝关节周围Campanacci Ⅲ级骨巨细胞瘤患者采用肿瘤整块切除,酒精灭活瘤段骨复合假体重建,男5例,女3例;年龄20~40岁,平均31岁。股骨远端5例,胫骨近端3例。4例为原发肿瘤、余4例为复发病例,其中2例伴有病理性骨折。术后根据国际骨肿瘤协会(Musculoskeletal Tumor Society, MSTS)“骨与软组织肿瘤术后功能重建的评估标准”进行肢体功能评估。根据国际保肢学会(International Society of Limb Salvage,ISOLS)影像评分标准(复合移植)进行影像学评估。结果 随访38~67个月,平均54个月,未出现复发、转移、假体松动等。灭活骨与宿主骨平均愈合时间5.5个月。随访末期,MSTS评分为25~29分,平均为26.3分(88%),ISOLS复合移植评分为28~35分,平均为32.8分 (88.5%)。1例术后3个月局部疼痛,X线片显示宿主骨-灭活骨有轻微骨吸收,非负重功能锻炼,术后6个月复查骨吸收消失。“爬行替代”可能是酒精灭活骨复合假体重建的主要骨愈合方式,股骨病例的愈合时间略快于胫骨病例。结论 对于膝关节周围Campanacci Ⅲ级骨巨细胞瘤,采用整块切除并酒精灭活瘤段骨复合假体重建可以取得满意的临床效果。     

Metachronous multicentric giant cell tumor: a case report and literature review

Metachronous multicentric giant cell tumors of bone are rare. The case of a 47-year-old woman who had a giant cell tumor of the ilium develop 24 years after resection of a giant cell tumor of the proximal tibia is reported. The initial and current surgical approaches for this patient are described. A literature review is presented to show that this patient had the longest disease-free interval documented for a patient with metachronous multicentric giant cell tumors.     

Late development of malignant fibrous histiocytoma at the site of a giant cell tumour 38 years after initial surgery

The authors report on a patient who developed a malignant fibrous histiocytoma at the site of a benign giant cell tumour, which had been treated by curettage 38 years previously. This latency period is, to their knowledge, the longest yet reported. This female patient was initially treated for a benign giant cell tumour of the proximal tibia when she was 33 years old; she underwent curettage and Kiel bone grafting. She had not received radiation therapy. Twenty eight years later, she underwent a second operation due to recurrence of a tumour. No specific histological diagnosis was possible: histology suggested a benign tumour, however compatible with a low-grade malignant potential but not associated with giant cell tumour. The patient underwent a third operation, with extensive curettage and total knee arthroplasty 38 years after the initial surgery, because of progressive knee pain. Postoperative histopathology study showed high-grade malignant fibrous histiocytoma. Finally, she underwent above-knee amputation because of uncontrollable progression of the tumour. The use of xenogenic bone graft, bone cement and associated bone necrosis potentially contributed to the development of a malignant tumour adjacent to the primary giant cell tumour.     

Case Report: Reconstruction of the Distal Tibia with Porous Tantalum Spacer after Resection for Giant Cell Tumor

Treatment options for giant cell tumors of the distal tibia include curettage and cement packing, curettage and bone grafting, or resection and reconstruction for aggressive tumors. Curettage of aggressive tumors often leads to severe bone loss requiring reconstruction. Allograft and autograft may be effective options for reconstruction, but each is associated with drawbacks including the possibility of infection and collapse. We present a case of giant cell tumor of the distal tibia treated with curettage and arthrodesis using a porous tantalum spacer. Complete removal of the tumor and successful arthrodesis of the ankle were accomplished using the spacer. The patient returned to pain-free walking along with eradication of the giant cell tumor. We believe porous tantalum spacers are a reasonable option for reconstructing the distal tibia after curettage of a giant cell tumor with extensive bone loss.     

吻合血管腓骨髂骨联合移植修复膝关节周围骨巨细胞瘤切除术后的骨缺损

目的探讨吻合血管的腓骨、髂骨联合移植修复膝关节周围骨巨细胞瘤扩大切除的方法及疗效。方法1996年10月~2002年11月,收治膝关节周围骨巨细胞瘤25例,其中17例采用肿瘤扩大切除,吻合血管的腓骨、髂骨联合移植术,年龄18~44岁。Enneking分期均为A期;放射影像学Campanicci’s分级级11例,级5例,级1例;病理学Jaffe's分级级9例,级7例,级1例。骨缺损范围为5.0cm×4.0cm~8.0cm×5.5cm,采用胫骨近端肿瘤扩大切除吻合血管的腓骨、髂骨联合移植修复肿瘤切除后骨缺损9例,股骨远端肿瘤扩大切除吻合血管的腓骨、髂骨联合移植修复重建肿瘤切除后骨缺损8例。随访观察术后植骨愈合、肿瘤复发情况、膝关节功能以及死亡情况。结果术后17例移植髂骨4.0cm×4.0cm~5.0cm×5.0cm,移植腓骨长度14~20cm,伤口期愈合。均获随访26~87个月,平均54个月。所有移植骨术后均愈合,愈合时间75~120d,平均93d。术后复发2例,关节腔间隙轻度狭窄3例。所有患者膝关节屈伸功能良好,其中股骨远端重建后屈曲80~105°,平均96°;胫骨近端重建后屈曲90~120°,平均110°。根据Enneking下肢功能评价标准,优11例,良3例,可1例,差2例,优良率为82.4%。结论肿瘤扩大切除,应用吻合血管的腓骨、髂骨联合移植修复膝关节周围骨巨细胞瘤具有肿瘤切除相对彻底、复发率低、骨移植愈合时间短、功能恢复快,能尽量保持和重建膝关节结构和功能等优点。     

Benign giant cell tumor of the spine: an unusual indication for radiotherapy

Introduction Giant cell tumors are rare primary bone tumors. Multicentricity as well as spinal localization is extremely rare.Case report We report about a 64-year-old female patient presenting with history of three osseous and one pulmonal manifestation of a benign giant cell tumor that have manifested metachronously within 23 years. The two periphery bone and the one pulmonal manifestation were treated surgically with success. Now, a further osseous lesion occurred at the seventh vertebra of the thoracic spine and was treated by dorsal instrumentation and replacement of the seventh vertebra. Nine months later, local recurrence of this benign giant cell tumor developed at the thoracic spine and was treated with radiotherapy with a total dose of 45 Gy. Three months after salvage radiotherapy definite local progress occurred localized within the former radiation treatment field. Due to neurological deficits a laminectomy and a stabilization of the destroyed sixth vertebra with bone cement was carried out. Histopathological examination again showed benign giant cell tumor without suspicion of malignancy.Conclusion In the literature the use of radiation therapy remains an appropriate therapy option in benign giant cell tumors with minimal adverse sequelae if primary surgical treatment is not feasible or fails. In cases of definitive radiotherapy a total dose > 45 Gy should be discussed.     

Giant cell tumors of the distal radius: resection: reconstruction by long avascular fibular graft (a case report)

Giant cell tumors (GCT) are locally aggressive tumors with a preference for epiphyses and metaphyses of long bones. They represent 5%-10% of all primary bone tumors. They affect mostly young adults between 20 and 40. Their origin remains uncertain. GCT is a purely lytic tumor, recurrent and can even lead to fracture. The distal radius is the third location after the distal femur and proximal tibia. Tumors are benign on histopathology, but "benign" lung metastases can sometimes be seen. Their treatment remains controversial because of the high rate of recurrence; oncological resection of the diseased bone segment with reconstruction reduces the rate of recurrence. Several techniques of resection and reconstruction of the wrist have been proposed. We report a case of giant cell tumor of the distal radius treated by resection and reconstruction by avascular fibular graft to a length of 12cm, and we evaluate the use of this reconstruction to salvage the wrist with this pathology.     

Reconstruction by bone transport after resection of benign tumors of tibia: A retrospective study of 38 patients

Background:

The commonly used reconstructive options after post resection defects in bone tumors like megaprosthesis, autograft, allograft, bone graft substitutes and recycled bone have their own demerits on a long term. Bone transport that regenerates patient''s own bone is a less explored option of reconstruction after resection of benign bone tumors and reports on this are limited. This technique is very much relevant in tibia where Ilizarov fixator is surgeon and patient friendly. We report our experience.

Materials and Methods:

This is a retrospective series of resection and bone transport in 38 patients with benign tumor of tibia. There were 14 males and 24 females with mean age of 23.40 years (range 9–40 years). Lesion was located in proximal third tibia in 27, middle third in two and distal third in nine patients. The diagnosis was giant cell tumor in 32, chondroblastoma in three, chondromyxoid fibroma, enchondroma and desmoplasic fibroma in one patient each. The resection was intercalary in 28 and transarticular in 10 patients. Osteosynthesis was monofocal in three, bifocal in 31 and polyfocal in four cases.

Results:

Mean followup was 7.22 years (range 1.5–15 years). Mean resection length was 10.21 cm (range 3–22 cm). The mean duration of external fixator was 308.03 days (range 89–677 days) and mean external fixator index was 36.14 days/cm (range 16.84–97.43 days/cm). Twelve patients had difficulties in the form of 11 problems and five obstacles that were successfully managed. None of the patients had local recurrence of tumor or any long term complication. Mean Musculo-skeletal Tumour Society score at final followup was 27.18 (90.60%).

Conclusions:

Bone transport is an excellent option after resection of benign tumors of tibia with good local control and functional outcome, despite minor difficulties that need timely management.     

Giant cell tumor of distal phalanx of great toe. A case report

Giant cell tumors are locally aggressive benign osseous neoplasms of unknown origin. They mostly occur after skeletal maturity in 3rd or 4th decade and commonly involve long bones although occasional occurrence at other sites has been reported. It is rare to see these tumors involving the phalangeal bone of foot. We report a case of giant cell tumor involving the distal phalanx of great toe in a 27 year old female who presented with swelling of great toe of right foot. Radiography showed an expansile lesion in distal phalanx of great toe. En bloc resection of phalanx was done. Biopsy showed giant cell tumor and regular follow up of this patient for two years showed no recurrence of tumor. Giant cell tumor at such a location is unusually aggressive and needs regular follow up to detect local recurrence.     

Multicentric giant cell tumor of the fourth and fifth metacarpals with lung metastases

Giant cell tumors of bone (GCTB) are generally benign neoplasms, but recently, some authors consider them to be low-grade malignant neoplasms because they have a relatively high rate of recurrence and at least some potential for metastases. The majority of GCTB are unifocal, and less than 1 % are multicentric. We report a rare case of a multicentric GCTB arising simultaneously in the non-dominant fourth and fifth metacarpals of a 25-year-old female. The patient underwent ray amputation of the two involved digits, and the surgical margins were histologically negative for tumor. The tumor had the classic histologic appearance of a benign GCTB. A year after the amputation, the patient developed pulmonary metastasis which was treated with pulmonary lobe resection. She is currently over 2.5 years postsurgical treatment of the primary lesion with no evidence of local recurrence or distant metastasis.     

Primary pulmonary malignant meningioma.

Fewer than 20 cases of primary pulmonary meningioma have been reported. Most of these cases have been histologically and clinically benign. We report an unusual case of primary pulmonary malignant meningioma with atypical histologic features and malignant behavior. A computed tomography scan of the head did not show evidence of tumor. The right upper lobe mass was resected and showed features of an atypical meningioma with loss of architectural pattern, mild nuclear pleomorphism, increased mitotic counts (up to 15 mitotic figures per 10 high power fields), and focally prominent nucleoli. Focally, cells with rhabdoid features were identified. The tumor's immunohistochemical and ultrastructural profiles were consistent with a meningioma. The tumor stained negative for estrogen and focally positive for progesterone receptors and had a MIB-1 labeling index (marker of cell proliferation) of 9.2%. Approximately 5 months after the initial resection, the patient experienced a tumor recurrence with multiple lymph node metastases, spread to the middle and lower lobes of the right lung, and metastasis to the diaphragm. Rarely, primary pulmonary meningiomas may present as high-grade malignant lesions.     

外科治疗肢体骨巨细胞瘤43例

目的探讨外科手术治疗肢体骨巨细胞瘤（giant cell tumor of bone, GCT）的临床疗效。方法回顾性分析2007年1月至2013年7月于我院应用外科手术方法治疗的43例GCT患者,年龄20~66岁（平均32岁）;发病部位：股骨远端17例,胫骨近端16例,桡骨远端5例,腓骨近端2例,股骨近端2例,肱骨近端1例;治疗方法：病灶扩大刮除骨水泥填充并（或不并）内固定及并（或不并）植骨术,瘤段骨切除特制肿瘤关节置换术,瘤段骨切除腓骨小头移植术,瘤段骨切除关节融合术,瘤段骨切除稳定结构重建术。分析本组患者的手术相关资料、术后恢复情况及复发率。结果43例患者均获得随访,随访时间8~64个月,平均28个月。本组患者的总复发率为7.0%（3/43）,其中采用扩大刮除术患者复发率为7.7%（2/26）,瘤段切除术患者的复发率为5.9%（1/17）,复发患者均经二次手术治疗。2例合并感染,发生率为4.7%。结论通过术前认真设计,选择恰当的手术方式和重建方式,肢体GCT可获得良好的治疗效果。     

Clinical and immunohistochemical characteristics of benign giant cell tumour of bone with pulmonary metastases: case series

PURPOSE: Giant cell tumour of bone with pulmonary metastases is rare. However, some patients die of pulmonary metastases, and histological examination cannot distinguish between benign tumour and malignant metastases. In this study, we present clinical and immunohistochemical findings associated with giant cell tumour of bone with pulmonary metastases. METHODS: Five patients with benign giant cell tumour of bone with pulmonary metastases (one man and 4 women) were studied. Patients' ages ranged between 20 and 23 years (mean age, 21.8 years). Tumours were in the distal femur in 2 cases, and in the proximal tibia, distal tibia, and lumbar spine in one case each. The tissue specimens from primary tumours, recurrent tumours, and pulmonary metastases were studied using immunohistochemical techniques. RESULTS: Three of the 5 primary tumours were of the spontaneous regression or growth cessation type, or the continuously slow-growing type, showing 4.2% to 6.2% of positive cells for Ki-67 after immunohistochemical staining. However, 2 patients with the rapid-growing type of disease died of pulmonary metastases; their primary, recurrent, and metastatic tumour specimens contained 9.0% to 11.5% of positive cells for Ki-67. CONCLUSION: Three of the 5 primary tumours had a benign clinical pattern and immunohistochemistry. Two of the 5 patients died of pulmonary metastases, which had an aggressive clinical pattern and a high prevalence of positive cells in Ki-67. Examination of Ki-67 should be carried out for aggressive type of giant cell tumour.     

Histologically verified lung metastases in benign giant cell tumours—14 cases from a single institution

From 1975 to 1997, 649 cases of benign giant cell tumours of the bone were treated at the Istituto Rizzoli. Fourteen patients (2.1%) experienced lung metastases after a mean of 35.2 months. The time interval between the diagnosis and the appearance of the lung metastases ranged from 3 months to 11.9 years. Metastasectomy was performed in all patients. Histologically, the metastases were identical to the primary bone lesions. Two patients with unresectable multiple metastases received additional chemotherapy. After a follow-up of 70 months (range: 8.2 to 185 months), all patients are alive. Ten patients showed no evidence of disease, one of these after a second resection of metastases, and four patients presented stable disease with multiple lung metastases. Local recurrence of the bone lesion occurred in seven patients before or simultaneously to the metastases. In contrast to previous reports, we could not detect a predominance of the distal radius, but all of the patients had a stage III tumour according to the Enneking criteria of benign lesions. We conclude that even metastatic benign giant cell tumours have an excellent prognosis after adequate resection. No prognostic factors despite high-grade lesions were detectable.     

脊柱原发性肿瘤的切除方式与疗效分析

目的探讨脊柱原发性肿瘤分期指导切除方式的疗效。方法回顾性分析1997—2006年手术治疗的脊柱原发性肿瘤患者41例，男27例，女14例；年龄为1—71岁，平均30．5岁；良性23例（含嗜酸性肉芽肿11例），恶性18例（含骨巨细胞瘤7例）。结合Enneking和WBB分期分块切除肿瘤，椎体切除14例17个节段，后弓切除8例10个节段，全脊椎切除10例12个节段，其余类型11例。结果随访6—76个月，平均24个月。疼痛改善优良率97．6％。Frankel改善1级21例，改善2级7例，无改善13例。SF-36评分平均提高22．1。总复发率为12．2％（5／41），骨巨细胞瘤复发率为14．3％（1／7），恶性肿瘤复发率为36．4％（4／11）。结论Enneking和WBB分期指导脊柱原发性肿瘤手术，切除较彻底，近中期疗效良好，但仍存在缺陷，需进一步完善。SF-36可用于患者生命质量的全面评测。     

Long-term survival cases of lung cancer presented as solitary bone metastasis.

The prognosis of non-small cell lung carcinoma (NSCLC) with bone metastasis has been regarded as very poor. We report herein on two cases of NSCLC which presented as a solitary bone metastasis, were treated with surgical resection. Both these cases survived for over 5 years after their last operations. A 71-year-old-man was hospitalized with right crural pain. A diagnosis of squamous cell carcinoma of the left lower lobe with right fibula metastasis was made. A marginal resection of the right fibula was performed. After that, a left lower lobe lobectomy and systemic chemotherapy were carried out. He had a local recurrence in the right mediastinal lymph nodes eleven months after the operation. He received intraluminal and external radiation therapy and obtained complete remission. He has survived for 5 years without any other recurrence or metastasis. A 52-year-old-man was admitted to our hospital with left thigh pain. A diagnosis of adenocarcinoma of the right upper lobe with left thigh metastasis was made. A right upper lobe lobectomy and a resection of the left thigh tumor were performed. Three cycles of systemic chemotherapy were given after that. He has survived for 5 years since his last operation without any recurrence or metastasis.