Surgical treatment of giant cell tumours of the thoracic and lumbar spine: report of nine patients

Giant cell tumours involving vertebral bodies are still difficult to treat, though results are gradually improving. The object of this study was to assess the results of "complete excision", both of previously untreated giant cell tumours and of recurrences, and to consider the possible effects of any tumour contamination during operation. Nine consecutive patients with giant cell tumours of the thoracic and lumbar spine were treated surgically between 1986 and 1995. Four of these patients were referred with recurrent tumours. All operations aimed at complete resection of the tumour, where possible an en-bloc approach was used. The spines were reconstructed with autografts and instrumentation. All patients were regularly reviewed as part of an on-going study. Following the five operations for previously untreated tumours ("primary" operations), there were no local recurrences, but one patient died of pulmonary metastases. One of the four patients operated upon for a recurrence developed a further recurrence, which was excised 2 1/2 years ago. It would seem that giant cell tumours of the thoracic and lumbar spine, including recurrences, should be treated by complete excision. The en-bloc approach is the safest technique. Where an intralesional component is unavoidable, total removal of the (pseudo)capsule should be ensured by preliminary extralesional dissection. Any tumour spill should be meticulously removed. The use of frozen sections to check resection margins is advisable.     

Heterotransplantation of the cultured cell from a human giant cell tumour of bone

Summary We have transplanted cultured cells derived from a human giant cell tumour of bone (G-1 cell) into immunologically suppressed mice. The resulting growths were morphologically and cytokinetically analyzed. We have obtained information on the cytokinetic influences of anti-cancer agents on heterotransplanted tumours using the double labelling method.Tumour formation was noted in 56 out of 76 mice, i.e. 76%. In 3 mice which had been kept under observation the large tumours led to death.The heterotransplanted tumours of G-1 cells had morphologically malignant and non-epithelial characteristics. The cells appeared to have undergone malignant change during their long period of cultivation.Using the double labelling method, 5-FU was found to prolong the DNA synthetic time and decrease the cell birth rate of G-1 cells in vivo. Accordingly, 5-FU is the most effective anti-cancer agent to G-1 cells.In order to improve the prognosis in a malignant bone tumour the most effective anti-cancer agent should be determined by testing on a heterotransplanted tumour derived from the patient.

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Résumé Les auteurs ont greffé des cellules cultivées à partir d'une tumeur osseuse à cellules géantes humaine (cellule G-1) sur des souris ayant subi une suppression immunitaire. Les tumeurs résultantes ont été analysées sur le plan morphologique et cytocinétique. Grâce à la technique du double marquage, on a obtenu des informations concernant les influences cytocinétiques des agents anticancéreux sur les tumeurs hétérotransplantées.Des tumeurs se sont développées chez 56 souris sur 76, soit 76%. Chez trois animaux soumis à une observation prolongée, on a constaté que les volumineuses tumeurs qui se sont formées ont entraîné la mort.Les tumeurs hétérotransplantées des cellules G-1 étaient morphologiquement malignes et non épithéliales. Les cellules ont apparemment subi une transformation maligne pendant leur longue période de culture.A l'aide du double marquage on a trouvé que le 5-fluoro-uracile prolongeait le temps de synthèse de l'ADN tandis qu'il diminuait le taux de division des cellules G-1 in vitro. Le 5-fluoro-uracile est donc l'agent anticancéreux le plus efficace à l'égard des cellules G-1.Pour améliorer le pronostic d'une tumeur osseuse maligne, on devrait déterminer l'agent anticancéreux le plus efficace en le testant sur une tumeur hétérotransplantée dérivée du malade.

     

Tissue culture studies on human giant cell tumour and osteosarcoma

Summary Benign giant cell tumours showed a sharp chromosome number mode at 46 with a superficially normal diploid karyotype. Osteosarcomas, however, were characterised by having aneuploid modal numbers. LDH isozyme patterns of giant cell tumours showed a maximum activity in Fractions IV and V, whereas that of osteosarcomas apparently fell in a range of Fraction III.Thirty-three giant cell tumours and 22 osteosarcomas were cultured in vitro. In general, frequencies of polyploid and aneuploid cells increased with the accretion of in vitro generations, and LDH isozyme patterns showed a marked increased activity of Fraction V in the early subculture passages. Two permanent cell lines of giant cell tumours were successfully established; they showed striking morphological changes of cells and the modal chromosome number fell in a range of hypotriploidy. LDH isozyme pattern showed maximum activity in Fractions III and IV, respectively. An interesting fact is that these cell lines in later subculture generations, formed tumours by subcutaneous inoculations and tumour death occurred by intravenous inoculation. Histologically, the tumour nodules provided evidence for the presence of sarcomatous stromal cells. Furthermore, osteoid and chondroid tissues surrounded by sarcomatous cells occurred in subcutaneous tumours.

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Résumé Les tumeurs bénignes à cellules géantes ont un pic de fréquence chromosomique de 46 avec un caryotype diploïde apparemment normal. Par contre, les ostéosarcomes sont caractérisés par un pic de fréquence chromosomique aneuploïde. Les profils des isozymes de la lacticodéshydrogénase ont montré une activité maximum dans les fractions IV et V, tandis que celle des ostéosarcomes semblait appartenir à la Fraction III.Trente trois tumeurs à cellules géantes ainsi que vingt deux ostéosarcomes ont été cultivés in vitro. En général, la fréquence des cellules polyploïdes et aneuploïdes a augmenté au fur et à mesure des générations in vitro et les profils des isozymes de la lacticodéshydrogénase ont montré une nette augmentation de l'activité de la Fraction V pendant les transferts initiaux. Deux lignées permanentes de tumeurs à cellules géantes ont été établies. Ces cellules ont subi des changements morphologiques frappants et leur pic de fréquence chromosomique était dans la gamme d'une hypotriploïdie. L'activité maximum de la lacticodéshydrogénase des deux lignées a été retrouvée respectivement dans les Fractions III et IV.Il est intéressant de noter que, lors des générations ultérieures in vitro, ces lignées ont été capables de former des tumeurs par inoculation sous-cutanée et de provoquer la mort à la suite d'inoculation intraveineuse. Du point de vue histologique, les nodules tumoraux montraient la présence d'un stroma de cellules sarcomateuses. Par ailleurs, les tumeurs sous-cutanées étaient faites de tissu ostéoïde et chondroïde entouré de cellules sarcomateuses.

     

An unusual giant cell tumour of bone

Summary This is a report of an 18-year old girl with an unusual giant cell tumour. Although it is difficult to distinguish between a multifocal giant cell tumour of bone and a primary giant cell tumour with metastases in other bones, we believe that our case is either a multicentric or a multifocal giant cell tumour.

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Résumé Présentation d'une tumeur à cellules géantes de type inhabituel chez une jeune fille de 18 ans. Bien qu'il soit difficile de distinguer une tumeur à cellules géantes à foyers multiples d'une tumeur primitive accompagnée de métastases au niveau d'autres os, il semble bien que ce cas soit une tumeur à cellules géantes multifocale.

     

Multicentric metachronous giant cell tumour of vertebrae and tibia

Background Multiple metachronous giant cell tumours (GCT), occurring in an individual are extremely rare. Purpose A patient presenting with GCT of multiple thoracic and lumbar vertebrae, 14 year subsequent to occurrence of GCT of proximal end of left tibia, is being reported. Study design Case report. Method Forty-year-old male presented with upper motor neuron paraplegia with sphincter involvement and complete sensory involvement below T11 spinal level. There was a surgical scar of resection arthrodesis of left knee for GCT proximal tibia. Radiographs showed compression of T8, T10, T12, L1 and L4 vertebrae. Routine haematological investigations including serum PTH levels were normal. MRI scan and CT scan showed expansile mass lesion and compression of cord at D12 and L1 vertebrae. Posterior decompression and stabilization was performed. Results As the second focus of the disease occurred 14 years after the appearance of initial lesion, the second independent focus of the disease, rather than a metastasis, is the likely cause. Conclusion It is the first reported case of multiple giant cell tumour of the spine above the sacrum.     

Distal radioulnar joint prosthesis for the treatment of giant cell tumor of the distal ulna: a case report and literature review

Giant cell tumor (GCT) of the distal end of the ulna is an uncommon site for primary bone tumors. When it occurs, en-bloc resection of the distal part of the ulna with or without reconstruction stabilization of the ulnar stump is the recommended treatment. We present a case of a 56-year-old man with a GCT of the distal ulna treated successfully with an en-bloc resection of the distal ulna with reconstruction using radioulnar joint prosthesis. Although the experience with this type of treatment is limited, implantation of a metallic prosthesis to replace the distal part of the ulna can also be considered as a salvage procedure for the treatment of this difficult pathology.     

The dilemma of denosumab: Salvage of a femoral head giant cell tumour

INTRODUCTION

Denosumab is a monoclonal RANKL antibody which has been shown to be highly effective in treating giant cell tumour (GCT) of bone. We report on its use as a neo-adjuvant agent to avoid morbid surgery for an adolescent.

PRESENTATION OF CASE

We report a case of a15-year old female with a Campanacci 3 GCT involving the femoral head and neck.

DISCUSSION

To preserve bone stock and avoid an outright hip replacement, the patient was given denosumab pre-operatively to consolidate the tumour. After receiving 6 months of treatment, a rim of cortical bone had developed to allow an extended curettage of the tumour to be performed without fear of collapse of the articular surface.

CONCLUSION

This is the first reported case of the use of denosumab in GCT of the femoral head and neck. We describe our experience in the neo-adjuvant use of denosumab and offer suggestions for future use. Further studies will be needed to see if denosumab has a role in conventional GCT and whether it can lead to a lowering of local recurrence rates.     

The management of soft tissue tumours

Summary Malignancy must be suspected in any mass of uncertain diagnosis in the trunk or limbs. The history and clinical, and radiological examination, including angiography, will facilitate a reasonable presumptive diagnosis. Biopsy should only be undertaken by the surgeon who will perform the definitive operation and should be planned so that the biopsy route can be excised later with the tumour. Extirpation biopsy can be performed if conditions are favourable.The value of angiography as a diagnostic aid is emphasized.Ten illustrative cases are reported.

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Résumé On doit soupçonner la malignité en présence de toute tumeur du tronc ou des membres de diagnostic incertain. L'histoire, l'examen clinique et radiologique, y compris l'angiographie, sont susceptibles d'aider à porter un diagnostic de présomption raisonnable. La biopsie doit être réalisée par le chirurgien qui exécutera la totalité du traitement et elle sera effectuée de telle sorte que la cicatrice puisse être excisée secondairement avec la tumeur. On pourra réaliser une biopsie-exérèse si les conditions sont favorables.L'auteur insiste sur l'intérêt de l'angiographie pour établir le diagnostic et présente 10 cas démonstratifs.

     

分离培养骨巨细胞瘤破骨样细胞的方法学研究

本文通过组织块贴壁法、机械分离法和酶消化法对骨巨细胞瘤的破骨样细胞进行分离、培养。通过比较,结果显示：酶消化法提纯程度最高,收获量较多;机械分离法居中,但操作简单,对原位杂交、免疫组织化学和骨吸收功能检测具有一定的应用价值;组织块培养法需要时间长,各细胞成分混杂,对研究各细胞成分之间的关系,有一定帮助     

A rare case of giant cell tumour arising from anterior cruciate ligament: Its diagnosis and management

Tenosynovial giant cell tumour is a locally aggressive tumour arising from the synovia of the fibrous tissue surrounding the joints, tendon sheaths and tendons. Areas of predilection are the hand, and in the case of synovial joints, the knee joint is particularly affected. We describe a rare case of an intra-articular localized tenosynovial giant cell tumour arising from the anterior cruciate ligament (ACL) in a 27 year male who presented with pain and giving way of his left knee without prior history of any trauma. Tests for internal derangement of knee were negative. MRI reported an ACL tear with a heterogeneous fibrous mass attached to the distal part, most probably an organized haematoma. It was decided to do a diagnostic arthroscopy before proceeding for ACL reconstruction. Arthroscopy revealed a purple coloured mass attached to distal part of ACL. The mass was removed piecemeal using an additional posterolateral portal. ACL was found intact. Histopathology reported it to be tenosynovial giant cell tumour. The patient was asymptomatic at each subsequent follow up. It is a rare diagnosis which presented as an ACL tear; in such suspected cases it is prudent to perform a diagnostic arthroscopy before going for ACL reconstruction.     

A case report. Tenosynovial giant cell tumour in the tendon of extensor digitorium longus

Soft tissue lesions found within the foot and ankle can vary in their origin. Tenosynovial giant-cell tumours (TSGCT) are quoted as rare in their presentation yet also reported to be the sixth most common benign lesion in the foot.This article presents a case report following the patient journey suffering with a TSGCT of the fourth toe. From initial consultation, to further investigation with advanced imaging to a final decision on surgical excision following consideration of conservative management.     

Partial wrist arthrodesis versus arthroplasty for distal radius giant cell tumours

Purpose

The purpose of this study was to evaluate the clinical efficacy of using the proximal fibular graft for partial wrist arthrodesis or arthroplasty after the resection of giant cell tumours of the distal radius.

Methods

Between February 2006 and August 2010, 14 patients (seven males, seven females; average age, 35.7 years) with grade II and III giant cell tumours of the distal radius were treated by tumour resection and autologous proximal fibular grafts to reconstruct the wrist in our hospital. Seven patients each were treated by wrist arthroplasty and partial wrist arthrodesis, and were followed up for 2.2–6.8 years (average, 3.9 years).

Results

All patients achieved primary healing. No tumour recurrence was observed during follow-up in any of the patients. No statistically significant difference in forearm rotation was observed between patients undergoing the two different treatments. However, wrist flexion-extension activities were significantly better and the wrist grip strengths were significantly worse in the arthroplasty group than in the arthrodesis group. The Musculoskeletal Tumour Society score did not significantly differ between the groups.

Conclusions

Overall, joint arthroplasty remains a favourable treatment with regard to the functional outcome for giant cell tumours of the distal radius; however, some of these patients may have a weaker grip strength. In comparison, partial wrist fusion appears to provide a durable and stable wrist with good long-term functional outcome.     

Giant cell tumour of the sacrum: a suggested algorithm for treatment

To investigate the outcome of our management of patients with giant cell tumour of the sacrum and draw lessons from this. A retrospective review of medical records and scans for all patients treated at our unit over the past 20 years with a giant cell tumour of the sacrum. Of the 517 patients treated at our unit for giant cell tumour over the past 20 years, only 9 (1.7%) had a giant cell tumour in the sacrum. Six were female, three male with a mean age of 34 (range 15–52). All, but two tumours involved the entire sacrum and there was only one purely distal to S3. The mean size was 10 cm and the most common symptom was back or buttock pain. Five had abnormal neurology at diagnosis, but only one presented with cauda equina syndrome. The first four patients were treated by curettage alone, but two patients had intraoperative cardiac arrests and although both survived all subsequent curettages were preceded by embolisation of the feeding vessels. Of the seven patients who had curettage, three developed local recurrence, but all were controlled with a combination of further embolisation, surgery or radiotherapy. One patient elected for treatment with radiotherapy and another had excision of the tumour distal to S3. All the patients are alive and only two patients have worse neurology than at presentation, one being impotent and one with stress incontinence. Three patients required spinopelvic fusion for sacral collapse. All patients are mobile and active at a follow-up between 2 and 21 years. Giant cell tumour of the sacrum can be controlled with conservative surgery rather than subtotal sacrectomy. The excision of small distal tumours is the preferred option, but for larger and more extensive tumours conservative management may well avoid morbidity whilst still controlling the tumour. Embolisation and curettage are the preferred first option with radiotherapy as a possible adjunct. Spinopelvic fusion may be needed when the sacrum collapses.     

原位微波灭活瘤段＋重建治疗骨巨细胞瘤的临床观察

目的应用微波原位灭活瘤段＋重建技术治疗骨巨细胞瘤,观察临床疗效和肢体功能。方法 2004年3月至2011年4月本组共应用此方法治疗32例骨巨细胞瘤患者,其中骨盆1例,股骨远端12例,胫骨近端10例,桡骨远端4例,肱骨近端3例,肱骨远端1例,跟骨1例。病理分级：Ⅰ级4例,Ⅱ级11例,Ⅲ级17例。术中边缘外分离肿瘤,原位插入微波针,控制性高温灭活瘤体,自体骨粒或骨水泥填充病灶腔隙,填充骨水泥和异体骨混合物,部分需行内固定,观察术后肿瘤复发和肢体功能评分。结果 32例患者皆得到随访,平均随访2年8个月。发现1例复发,1例感染,肢体关节功能评定：好者28例,可者4例,差者0例。结论原位微波灭活瘤段＋重建技术治疗骨巨细胞瘤可能降低了骨巨细胞瘤术后的复发,并保留较好的肢体功能。     

The role of temporal artery biopsies in giant cell arteritis

A knowledge of the disease process of giant cell arteritis and its diagnosis can help a surgeon to decide which patients will benefit from a biopsy being performed and identify where a biopsy would be of no value in their management. This article discusses the issues involved.     

关节镜下手术治疗膝关节局灶型腱鞘巨细胞瘤

目的探讨膝关节内局灶型腱鞘巨细胞瘤的诊疗技术。方法1989年至2003年收治膝关节局灶型腱鞘巨细胞瘤患者12例,术前误诊半月板损伤6例,膝关节慢性滑膜炎3例,仅有3例通过MRI检查获得确诊。全部病例均采用关节镜下肿瘤完整切除术。结果9例患者获得1～10年随访,临床症状消失,膝关节活动正常,X线检查未见异常,未见复发病例。结论膝关节镜下手术治疗腱鞘巨细胞瘤是较佳方法,术中行瘤组织周围扩大切除及肿瘤取出组织通道处刨削清理,是防止肿瘤复发的关键所在。     

Osteoclastic giant cell tumor of the pancreas

INTRODUCTION

Pancreatic giant cell tumors are rare, with an incidence of less than 1% of all pancreatic tumors. Osteoclastic giant cell tumor (OGCT) of the pancreas is one of the three types of PGCT, which are now classified as undifferentiated carcinoma with osteoclast-like giant cells.

PRESENTATION OF CASE

The patient is a 57 year old woman who presented with a 3 week history of epigastric pain and a palpable abdominal mass. Imaging studies revealed an 18 cm × 15 cm soft tissue mass with cystic components which involved the pancreas, stomach and spleen. Exploratory laparotomy with distal pancreatectomy, partial gastrectomy and splenectomy was performed. Histology revealed undifferentiated pancreatic carcinoma with osteoclast-like giant cells with production of osteoid and glandular elements.

DISCUSSION

OGCT of the pancreas resembles benign-appearing giant cell tumors of bone, and contain osteoclastic-like multinucleated cells and mononuclear cells. OGCTs display a less aggressive course with slow metastasis and lymph node spread compared to pancreatic adenocarcinoma. Due to the rarity of the cancer, there is a lack of prospective studies on treatment options. Surgical en-bloc resection is currently considered first line treatment. The role of adjuvant therapy with radiotherapy or chemotherapy has not been established.

CONCLUSION

Pancreatic giant cell tumors are rare pancreatic neoplasms with unique clinical and pathological characteristics. Osteoclastic giant cell tumors are the most favorable sub-type. Surgical en bloc resection is the first line treatment. Long-term follow-up of patients with these tumors is essential to compile a body of literature to help guide treatment.     

Risk factors and oncological outcomes of pulmonary metastasis in patients with giant cell tumor of bone

BackgroundGiant cell tumor (GCT) of bone has a rare potential for metastatic spread. This study aimed at evaluating the incidence of chest metastases in GCT and their oncological outcome and identifying possible risk factors.MethodsMedical records of 466 (313 de novo and 153 recurrent) patients with primary GCT of bone were retrospectively reviewed. Fifteen (3.2%) patients developed chest metastasis. Time from diagnosis of the primary bone lesion to the diagnosis of metastasis, treatment modalities of metastasis, and the course of treatment were revised. The functional outcome was evaluated using the Musculoskeletal Tumor Society (MSTS) scoring system, and postoperative complications were recorded.ResultsThis study included 7 males and 8 females with a mean age of 27.3 ± 7.9 years. The most common site of the primary tumor was the distal femur. All fifteen patients were recurrent cases. The mean follow-up period was 67.7 ± 33.2 months. Chest metastasis was diagnosed after a mean time of 28.1 ± 28.9 months from the initial diagnosis of the bone lesion. One patient died of disease (DOD) 18 months after the surgical intervention. The incidence of chest metastasis in recurrent cases was 9.8%, while de novo cases did not develop chest metastasis, P < 0.001. Previous curettage was associated with a higher incidence of chest metastasis (14.6%) compared to previous resection (4.2%), P = 0.03.ConclusionsChest metastasis following GCT of bone is rare. Risk factors include recurrent cases, especially following previous curettage. Patients have a good prognosis and a low mortality rate.Level of evidenceLevel IV, retrospective.