The age-related impact of surviving sarcoma on health-related quality of life: data from the SURVSARC study.

BACKGROUND: Health-related quality of life (HRQoL) data of sarcoma survivors are scarce and the impact of age remains unclear. The aims of this population-based study were to (i) compare HRQoL scores amongst three age-groups [adolescents and young adults (AYA, aged 18-39 years), older adults (OA, aged 40-69 years) and elderly (aged ≥70 years)]; (ii) compare HRQoL of each sarcoma survivor age group with an age- and sex-matched normative population sample; (iii) determine factors associated with low HRQoL per age group. METHODS: Dutch sarcoma survivors, who were 2-10 years after diagnosis, were invited to complete the European Organization for Research and Treatment of Cancer Quality of Life Questionnaire-Core 30-questions questionnaire on HRQoL. RESULTS: In total, 1099 survivors (58% response rate) completed the questionnaire: 186 AYAs, 748 OAs and 165 elderly. The median time since diagnosis for all patients was 5.2 years. Bone sarcomas were seen in 41% of AYAs, 22% of OAs and in 16% of elderly survivors (P < 0.01). AYA and OA survivors reported statistically significant and clinically meaningful worse physical, role, cognitive, emotional and social functioning compared with a matched norm population, which was not the case for elderly survivors. AYAs reported significantly worse scores on emotional and cognitive functioning compared with OA and elderly survivors. Malignant peripheral nerve sheath tumour, osteosarcoma and chordoma were the subtypes of which survivors reported the lowest HRQoL scores in comparison with the norm. For all age groups, chemotherapy, having a bone sarcoma and having comorbidities were most frequently associated with low scores on HRQoL subscales, whereas a shorter time since diagnosis was not. CONCLUSION: In this nationwide sarcoma survivorship study, the disease and its treatment had relatively more impact on the HRQoL of AYA and OA survivors than on elderly survivors. These results emphasise the need for personalised follow-up care that not only includes risk-adjusted care related to disease relapse, but also age-adjusted care.

Can patient-specific finite element models better predict fractures in metastatic bone disease than experienced clinicians?: Towards computational modelling in daily clinical practice.

OBJECTIVES: In this prospective cohort study, we investigated whether patient-specific finite element (FE) models can identify patients at risk of a pathological femoral fracture resulting from metastatic bone disease, and compared these FE predictions with clinical assessments by experienced clinicians. METHODS: A total of 39 patients with non-fractured femoral metastatic lesions who were irradiated for pain were included from three radiotherapy institutes. During follow-up, nine pathological fractures occurred in seven patients. Quantitative CT-based FE models were generated for all patients. Femoral failure load was calculated and compared between the fractured and non-fractured femurs. Due to inter-scanner differences, patients were analyzed separately for the three institutes. In addition, the FE-based predictions were compared with fracture risk assessments by experienced clinicians. RESULTS: In institute 1, median failure load was significantly lower for patients who sustained a fracture than for patients with no fractures. In institutes 2 and 3, the number of patients with a fracture was too low to make a clear distinction. Fracture locations were well predicted by the FE model when compared with post-fracture radiographs. The FE model was more accurate in identifying patients with a high fracture risk compared with experienced clinicians, with a sensitivity of 89% versus 0% to 33% for clinical assessments. Specificity was 79% for the FE models versus 84% to 95% for clinical assessments. CONCLUSION: FE models can be a valuable tool to improve clinical fracture risk predictions in metastatic bone disease. Future work in a larger patient population should confirm the higher predictive power of FE models compared with current clinical guidelines.Cite this article: F. Eggermont, L. C. Derikx, N. Verdonschot, I. C. M. van der Geest, M. A. A. de Jong, A. Snyers, Y. M. van der Linden, E. Tanck. Can patient-specific finite element models better predict fractures in metastatic bone disease than experienced clinicians? Towards computational modelling in daily clinical practice. Bone Joint Res 2018;7:430-439. DOI: 10.1302/2046-3758.76.BJR-2017-0325.R2.

Cryosurgery as Additional Treatment in Tenosynovial Giant Cell Tumors.

Introduction. Tenosynovial giant cell tumors (TGCT) emerge from the synovium and can behave aggressively. Surgical resection is the standard treatment. However, up to half of the patients with diffuse type show recurrences. Several additional treatments have been applied to reduce recurrences; none of these treatments was proven to be superior to surgical resection solely. This article describes the results of additional cryosurgery to surgical resection. Materials and Methods. We retrospectively evaluated 141 TGCT patients, between 1999 and 2007. Twelve patients had additional cryosurgery. The knee (n = 8), hip (n = 2), ankle (n = 1), and elbow (n = 1) were affected. Primary outcome variables were treatment indications, recurrences, and complications. Results. Indications for additional cryosurgery were extended disease, bone involvement, and locations that are difficult to surgically get disease-free such as cruciate ligaments. Five patients had recurrent disease, all of which had prior treatments. None of the primary treated patients had recurrent disease. One patient had a deep infection. Discussion. Cryosurgery may serve as an additional treatment for diffuse TCGT in selected cases. However, because of the small number of patients and the heterogeneous group we could not prove an advantage of additional cryosurgery over surgical resection only. Cryosurgery should be considered for further evaluation in a prospective study. If there is any effect it would be helpful, especially in patients with multiple TGCT recurrences.

Hemicortical resection and inlay allograft reconstruction for primary bone tumors: a retrospective evaluation in the Netherlands and review of the literature.

BACKGROUND: Selected primary tumors of the long bones can be adequately treated with hemicortical resection, allowing for optimal function without compromising the oncological outcome. Allografts can be used to reconstruct the defect. As there is a lack of studies of larger populations with sufficient follow-up, little is known about the outcomes of these procedures. METHODS: In this nationwide retrospective study, all patients treated with hemicortical resection and allograft reconstruction for a primary bone tumor from 1989 to 2012 were evaluated for (1) mechanical complications and infection, (2) oncological outcome, and (3) failure or allograft survival. The minimum duration of follow-up was twenty-four months. RESULTS: The study included 111 patients with a median age of twenty-eight years (range, seven to seventy-three years). The predominant diagnoses were adamantinoma (n = 37; 33%) and parosteal osteosarcoma (n = 18; 16%). At the time of review, 104 patients (94%) were alive (median duration of follow-up, 6.7 years). Seven patients (6%) died, after a median of twenty-six months. Thirty-seven patients (33%) had non-oncological complications, with host bone fracture being the most common (n = 20, 18%); all healed uneventfully. Other complications included nonunion (n = 8; 7%), infection (n = 8; 7%), and allograft fracture (n = 3; 3%). Of ninety-seven patients with a malignant tumor, fifteen (15%) had residual or recurrent tumor and six (6%) had metastasis. The risk of complications and fractures increased with the extent of cortical resection. CONCLUSIONS: Survival of hemicortical allografts is excellent. Host bone fracture is the predominant complication; however, none of these fractures necessitated allograft removal in our series. The extent of resection is the most important risk factor for complications. Hemicortical resection is not recommended for high-grade lesions; however, it may be superior to segmental resection for treatment of carefully selected tumors, provided that it is possible to obtain adequate margins. LEVEL OF EVIDENCE: Therapeutic Level IV. See Instructions for Authors for a complete description of levels of evidence.

Giant cell tumors of the sacrum--a nationwide study on midterm results in 26 patients after intralesional excision.

PURPOSE: Evaluation of recurrences, complications and function at mid-term follow-up after curettage for sacral giant cell tumor (GCT). METHODS: We retrospectively studied all 26 patients treated for sacral GCT in the Netherlands (from 1990 to 2010). Median follow-up was 98 (6-229) months. All patients underwent intralesional excision, 21 with local adjuvants, 5 radiotherapy, 3 IFN-α, 1 bisphosphonates. Functional outcome was assessed using Musculoskeletal Tumor Society (MSTS) score. Statistics were performed with Kaplan-Meier, Cox regression, log rank and Mann-Whitney U. RESULTS: Recurrence rate was 14/26 after median 13 (3-139) months and was highest after isolated curettage (4/5). Soft tissue masses >10 cm increased recurrence risk (HR = 3.3, 95 % CI = 0.81-13, p = 0.09). Complications were reported in 12/26 patients. MSTS was superior in patients without complications (27 vs. 21; p = 0.024). CONCLUSION: Recurrence rate for sacral GCT was highest after isolated curettage, indicating that (local) adjuvant treatment is desired to obtain immediate local control. Complications were common and impaired function.

Intercalary allograft reconstructions following resection of primary bone tumors: a nationwide multicenter study.

BACKGROUND: Favorable reports on the use of massive allografts to reconstruct intercalary defects underline their place in limb-salvage surgery. However, little is known about optimal indications as reports on failure and complication rates in larger populations remain scarce. We evaluated the incidence of and risk factors for failure and complications, time to full weight-bearing, and optimal fixation methods for intercalary allografts after tumor resection. METHODS: A retrospective study was performed in all four centers of orthopaedic oncology in the Netherlands. All consecutive patients reconstructed with intercalary (whole-circumference) allografts after tumor resection in the long bones during 1989 to 2009 were evaluated. The minimum follow-up was twenty-four months. Eighty-seven patients with a median age of seventeen years (range, 1.5 to 77.5 years) matched inclusion criteria. The most common diagnoses were osteosarcoma, Ewing sarcoma, adamantinoma, and chondrosarcoma. The median follow-up period was eighty-four months (range, twenty-five to 262 months). Ninety percent of tumors were localized in the femur or the tibia. RESULTS: Fifteen percent of our patients experienced a graft-related failure. The major complications were nonunion (40%), fracture (29%), and infection (14%). Complications occurred in 76% of patients and reoperations were necessary in 70% of patients. The median time to the latest complication was thirty-two months (range, zero to 200 months). The median time to full weight-bearing was nine months (range, one to eighty months). Fifteen grafts failed, twelve of which failed in the first four years. None of the thirty-four tibial reconstructions failed. Reconstruction site, patient age, allograft length, nail-only fixation, and non-bridging osteosynthesis were the most important risk factors for complications. Adjuvant chemotherapy and irradiation had no effects on complication rates. CONCLUSIONS: We report high complication rates and considerable failure rates for the use of intercalary allografts; complications primarily occurred in the first years after surgery, but some occurred much later after surgery. To reduce the number of failures, we recommend reconsidering the use of allografts for reconstructions of defects that are ≥15 cm, especially in older patients, and applying bridging osteosynthesis with use of plate fixation.

Giant cell tumours of the small bones of the hands and feet: long-term results of 30 patients and a systematic literature review.

Giant cell tumours (GCTs) of the small bones of the hands and feet are rare. Small case series have been published but there is no consensus about ideal treatment. We performed a systematic review, initially screening 775 titles, and included 12 papers comprising 91 patients with GCT of the small bones of the hands and feet. The rate of recurrence across these publications was found to be 72% (18 of 25) in those treated with isolated curettage, 13% (2 of 15) in those treated with curettage plus adjuvants, 15% (6 of 41) in those treated by resection and 10% (1 of 10) in those treated by amputation. We then retrospectively analysed 30 patients treated for GCT of the small bones of the hands and feet between 1987 and 2010 in five specialised centres. The primary treatment was curettage in six, curettage with adjuvants (phenol or liquid nitrogen with or without polymethylmethacrylate (PMMA)) in 18 and resection in six. We evaluated the rate of complications and recurrence as well as the factors that influenced their functional outcome. At a mean follow-up of 7.9 years (2 to 26) the rate of recurrence was 50% (n = 3) in those patients treated with isolated curettage, 22% (n = 4) in those treated with curettage plus adjuvants and 17% (n = 1) in those treated with resection (p = 0.404). The only complication was pain in one patient, which resolved after surgical removal of remnants of PMMA. We could not identify any individual factors associated with a higher rate of complications or recurrence. The mean post-operative Musculoskeletal Tumor Society scores were slightly higher after intra-lesional treatment including isolated curettage and curettage plus adjuvants (29 (20 to 30)) compared with resection (25 (15 to 30)) (p = 0.091). Repeated curettage with adjuvants eventually resulted in the cure for all patients and is therefore a reasonable treatment for both primary and recurrent GCT of the small bones of the hands and feet.

Aneurysmal bone cyst: the role of cryosurgery as local adjuvant treatment.

BACKGROUND: Aneurysmal bone cysts (ABCs) are most often treated with intralesional surgery (curettage) and additional bone grafting. There is debate on whether or not to use adjuvant therapy to decrease the local recurrence rate. This study is done to assess the outcome of curettage and cryosurgery as a treatment of ABC. METHODS: We analyzed 80 consecutive cases of ABC treated with curettage and cryosurgery. Patients were followed minimal 24 months after surgery (average 55 months, range 24-122 months) with physical examination and radiographs. Functional outcome was evaluated using the musculoskeletal tumor society score (MSTS). RESULTS: The 80 patients were all treated with curettage and cryosurgery. Additional bone grafting was used in 73 patients, osteosynthesis in 12 and spondylodesis in 1 patient. Four local recurrences were found in this study, a recurrence rate of 5%. All local recurrences were treated successfully with curettage and cryosurgery again 7-33 months after the initial surgery. Postoperative one fracture, one wound infection, and three transient nerve palsy occurred. The average MSTS score was 29.2 at follow-up. CONCLUSIONS: The use of cryosurgery as adjuvant therapy results in a lower local recurrence rate when compared to other publications for the treatment of ABC and excellent functional results.

Oncological and functional results of cryosurgical therapy of enchondromas and chondrosarcomas grade 1.

BACKGROUND AND OBJECTIVES: Cryosurgery using liquid nitrogen is used as adjuvant treatment after intralesional resection of bone tumours to induce cell death. It is applied to enlarge the oncological margins of resection and to reduce the local recurrence rate. The objective of this study is to analyze the oncological and functional results. METHODS: We studied the oncological and functional results of curettage and cryosurgery in 123 patients with 130 tumors. There were 75 enchondromas and 55 chondrosarcomas grade 1. The minimal follow-up period for all patients was 2 years (range 24-119 months). RESULTS: During follow-up one local recurrence of an active enchondroma and one local recurrence of an aggressive enchondroma occurred. They were treated with curettage and cryosurgery again. Both patients were disease-free at minimum of 3 years follow-up. No local recurrences after treatment of chondrosarcoma grade 1 were seen. Functional scores, according to the MSTS scoring system, showed an average score of 28 points (94%) at 2 years follow-up. Post-operative fractures were seen in 18 patients (14%). CONCLUSIONS: Curettage and cryosurgery for enchondroma and chondrosarcoma grade 1 has excellent oncological and functional results. The post-operative management has been adjusted to reduce the number of fractures.

Teardrop-femoral head distance after shelf acetabuloplasty for Perthes' disease.

Shelf acetabuloplasty is an accepted treatment for Perthes' disease. Favourable outcomes have been reported. This study aimed at determining whether changes in femoral head subluxation after shelf acetabuloplasty occur and whether they predict outcome. We assumed that reduction of subluxation after shelf acetabuloplasty would improve the long-term outcome. During follow-up, growth presented as a source of error when comparing the amount of absolute subluxation. Correction for growth was achieved by calculating a subluxation index. This index showed a significant reduction during follow-up. There was no correlation between clinical and radiological outcome, but the fact that the subluxation index showed a decreasing trend could be promising regarding outcome. This might be one of the reasons why shelf acetabuloplasty produces its effect. Following shelf acetabuloplasty the enlarged acetabulum could possibly direct the growth of the femoral head to a better-contained joint. This could result in better joint congruity and lesser tendency to degenerative arthritis.

Chondrosarcoma of bone: functional outcome and quality of life.

BACKGROUND AND OBJECTIVES: Outcome studies of chondrosarcoma have so far only reported oncological and functional results. Quality-of-life assessment becomes more important because more long-term survival after treatment of malignant diseases occur. The objective of this study is to analyze functional outcome and quality of life. METHODS: Functional evaluation of the patients was carried out according to the MSTS functional scoring system. For assessment of quality of life, questionnaires were used. RESULTS: Response of the questionnaires sent to 45 disease-free patients was 84%. The mean functional score of these 38 patients was 74% (20-100%). Best functional results were seen after bone graft reconstruction and curettage and cryosurgery with reconstruction. Quality-of-life analysis revealed a mean global health status of 75 (66-84). Furthermore, 24% of the patients experienced severe fatigue. Patients reported more problems with regard to physical functioning and sleep in comparison with healthy controls. No correlation was found between global health status scores and functional scores. CONCLUSIONS: Functional scores depend on the type of treatment with best results after curettage and cryosurgery. Quality-of-life analysis reveals problems on several domains, including fatigue, physical functioning and sleep.