LUMiC Endoprosthetic Reconstruction of Periacetabular Tumor Defects: A Multicenter Follow-up Study.

UPDATE: This article was updated on July 17, 2024 because of a previous error, which was discovered after the preliminary version of the article was posted online. The byline that had read "Richard E. Evenhuis, MD 1 , Michiel A.J. van de Sande, MD, PhD 1,2 , Marta Fiocco, PhD 2,3,4 , Demien Broekhuis, MD 1 , Michaël P.A. Bus, MD, PhD 1 , and the LUMiC® Study Group*" now reads "Richard E. Evenhuis, MD 1 , Michiel A.J. van de Sande, MD, PhD 1,2 , Marta Fiocco, PhD 2,3,4 , Edwin F. Dierselhuis, MD, PhD 5 , Demien Broekhuis, MD 1 , Michaël P.A. Bus, MD, PhD 1 , and the LUMiC® Study Group*". The Department of Orthopaedic Surgery, Radboudumc, Nijmegen, The Netherlands, has been added as the affiliation for Edwin F. Dierselhuis, MD, PhD. BACKGROUND: We previously reported promising early results for periacetabular tumor reconstructions using the LUMiC prosthesis. The current study evaluates mid-term complications, revision rates, cumulative incidence of implant revision, and risk factors for complications in a multicenter cohort. METHODS: We assessed patients in whom a tumor defect after type P1b+2, P2, P2+3, or P1b+2+3 internal hemipelvectomy was reconstructed with a LUMiC prosthesis during the period of 2008 to 2022. Complications were reported according to the Henderson classification. Competing risks models were used to estimate the cumulative incidence of implant revision for mechanical and nonmechanical reasons, and reoperations for any complication. Cox models were used to study the effect of risk factors on dislocation and infection. RESULTS: One hundred and sixty-six patients (median follow-up, 4.2 years [interquartile range, 2.6 to 7.6 years]) were included. A total of 114 (69%) were treated for a primary malignant tumor, 46 (28%) for metastatic carcinoma, 5 (3%) for a benign aggressive lesion, and 1 (1%) for another reason. One hundred and sixty-five reoperations were performed in 82 (49%) of the patients; 104 (63%) of the reoperations were within 6 months. Thirty-two (19%) of 166 implants were revised: 13 (8%) for mechanical reasons, mainly dislocation (n = 5, 3%), and 19 (11%) for nonmechanical reasons, mainly periprosthetic joint infection (PJI) (n = 15, 9%). The cumulative incidences of revision for mechanical reasons and PJI (Henderson 1 to 4) at 2, 5, and 10 years were 11% (95% confidence interval [CI], 7% to 17%), 18% (12% to 25%), and 24% (16% to 33%), respectively. Previous surgery at the same site was associated with an increased dislocation risk (cause-specific hazard ratio [HR CS ], 3.0 [95% CI, 1.5 to 6.4]; p < 0.01), and resections involving the P3 region were associated with an increased infection risk (HR CS , 2.5 [95% CI, 1.4 to 4.7]; p < 0.01). CONCLUSIONS: Despite a substantial reoperation risk, the LUMiC prosthesis demonstrated its durability in the mid-term, with a low mechanical revision rate and most patients retaining their primary implant. Most complications occur in the first postoperative months. Patients with previous surgery at the same site had an increased dislocation risk and might benefit from more conservative rehabilitation and aftercare. Measures should be aimed at reducing the PJI risk, especially in resections involving the P3 region. LEVEL OF EVIDENCE: Therapeutic Level IV . See Instructions for Authors for a complete description of levels of evidence.

Pediatric diaphragmatic hernia induced by a rib osteochondroma.

Diaphragmatic hernia in children is uncommon, especially when not congenital. We present a case of an 11-year old boy with a diaphragmatic hernia caused by a rib osteochondroma. The osteochondroma was surgically removed and the laceration in the diaphragm was repaired. This case shows the importance of being familiar with acquired diaphragmatic hernia in children, to recognize and prevent possible complications in an early stage.

Functional outcomes after reverse shoulder megaprosthesis following resection of malignant bone tumor in the proximal humerus: a systematic review and meta-analysis.

After resection of tumors in the proximal humerus, orthopedic oncologic surgeons are able to restore the shoulder function of patients with reverse shoulder megaprosthesis. Information about expected postoperative physical functioning is required to guide patient expectations, identify abnormal recovery, and set treatment goals. The aim was to provide an overview of functional outcomes after reverse shoulder megaprosthesis in patients after proximal humerus resection. This systematic review searched studies in MEDLINE, CINAHL, and Embase up to March 2022. Data on performance-based and patient-reported functional outcomes were extracted using standardized data extraction files. A meta-analysis with random effects model was performed to estimate outcomes after 2-year follow-up. The search identified 1089 studies. Nine studies were included in the qualitative analysis and six in the meta-analysis. Forward flexion range of motion (ROM) after 2 years was 105 degrees (95% Confidence Interval [CI]: 88-122, n = 59), abduction ROM 105 degrees (95% CI: 96-115, n = 29), and external rotation ROM 26 degrees (95% CI: 1-51, n = 48). The mean American Shoulder and Elbow Surgeons score after 2 years was 67 points (95% CI: 48-86, n = 42), mean Constant-Murley-Score 63 (95% CI: 62-64, n = 36), and mean Musculoskeletal Tumor Society score 78 (95% CI: 66-91, n = 56). The meta-analysis shows acceptable functional outcomes 2 years after reverse shoulder megaprosthesis. However, outcomes may well differ between patients as reflected by the CIs. Further research should focus on modifiable factors associated with impaired functional outcomes.

Active surveillance of atypical cartilaginous tumours of bone: short term quality of life measurements.

BACKGROUND: In the recent years active surveillance has been introduced for atypical cartilaginous tumours (ACT). This is the first study on the impact of this new treatment approach on patients' quality-of-life. We evaluated general health-related quality of life (HRQL) in patients diagnosed with enchondroma or ACT. METHODS: In this prospective study, patients recently diagnosed with enchondroma and ACT of the long bones were asked to participate. Health-related quality of life (HRQL) was assessed at diagnosis and at six month follow-up, using the 36-item Short Form Health Survey (SF-36) and Numeric Pain Rating Scale (NRS). HRQL of the active surveillance group was compared to the Dutch population and a Dutch sample with locoregional cancer. RESULTS: In total, 45 patients were included in the study, of which four patients underwent curettage and cryosurgery, 41 patients were under active surveillance. The HRQL of the active surveillance group seemed lower compared to the Dutch population, but similar to patients suffering locoregional cancers. No comparison between the surgery and the active surveillance group could be made. In the active surveillance group no statistical difference was found between baseline and six months follow-up regarding HRQL and pain during rest and activities. CONCLUSION: Patients diagnosed with non-malignant chondroid tumours have lower HRQL compared to the healthy population. Active surveillance had no adverse effect on patients well-being, after six months active surveillance the HRQL remained unchanged. Interestingly, in our study no impact on mental health was seen, implicating that diagnosed but untreated chondroid tumours do not seem to influence patients anxiety.

Shared decision making: Does a decision aid support patients with an atypical cartilaginous tumor in making a decision about treatment.

Objective: Due to new insights, atypical cartilaginous tumors (ACTs) of the long bones are no longer considered malignant and treatment is shifting from surgery to active surveillance. We developed a decision aid in order to support in shared decision making on treatment.The aim of this study is to evaluate the treatment preferences of patients with an ACT in the long bones. Methods: During thirty-four months, patients received a decision aid digitally with information about the disease, the treatment options, and the risks and benefits of active surveillance and surgical treatment. The given answers to patients' preference questions were evaluated qualitatively in relation to the final choice of treatment. Results: Eighty-four patients were included. None of the patients who preferred active surveillance later underwent surgery. Only four patients underwent surgery based on patient preference. Conclusion: In our experience the decision aid is useful for shared decision making as it provides the patient with information and the clinician with insight into patient's preferences. The preference for treatment generally corresponds to the eventual treatment. Innovation: When treatment changes, due to new insights, a decision aid seems helpful for both patients and clinicians to discuss the treatment that best suits the patient's situation.

Midterm MRI Follow-Up of Untreated Enchondroma and Atypical Cartilaginous Tumors in the Long Bones.

Management of atypical cartilaginous tumors (ACTs) in the long bones is shifting towards active surveillance to avoid unnecessary surgeries. The frequency and duration of active surveillance for these tumors is unclear as there is little knowledge of its biological behavior. In this retrospective study, we examined the natural course of enchondroma and ACTs through active surveillance. A total of 128 central cartilaginous tumors, located in the long bones, with a minimum interval of 24 months between baseline and last MRI were included. MRI characteristics (e.g., size, scalloping, fat entrapment) were scored and tumors were classified according to the changes between MRIs. Mean follow-up of this study was 50 months, range = 25-138 months. The majority of the cartilaginous tumors (87%) remained stable (n = 65) or showed regression (n = 46) on MRI. A total of 87% of the cases that developed tumor regression presented with entrapped fat at diagnosis. Only 13% (n= 17) showed some progression on MRI, although none of the tumors developed characteristics of high-grade chondrosarcoma. Based on our results, active surveillance is considered safe for enchondroma and ACTs of the long bones. We propose active surveillance for all asymptomatic enchondroma or ACTs in the long bones irrespective of tumor size, and follow-up schemes should be tailored on natural course.

Curettage and cryosurgery for enchondroma and atypical cartilaginous tumors of the long bones: Oncological results of a large series.

BACKGROUND AND OBJECTIVES: Intralesional surgical treatment is the preferred therapy for atypical cartilaginous tumors (ACTs) of the long bones in many institutions. However, the literature is still controversial regarding intralesional treatment versus wide resection. Due to the relative rarity of these tumors, studies reporting on the results of intralesional treatment are often small sample studies. METHODS: We retrospectively analyzed the oncological results of 55 enchondromas, 119 ACTs, and 5 chondrosarcomas grade 2 (CS2) treated with curettage and cryosurgery between the years 2004 and 2017 at our institution. The median follow-up period was 53 months (range, 24-169 months). RESULTS: In total, seven cases (three ACT, four CS2) recurred. Residual tumor was detected in 20 cases. Three cases underwent secondary curettage and cryosurgery due to local recurrence. Four cases underwent wide resection and reconstruction due to local recurrence with aggressive imaging characteristics. In total, 20 postoperative complications were seen. CONCLUSION: Curettage and cryosurgery for enchondroma and ACT show very good oncological results with a low recurrence rate and acceptable complication rate. Curettage and cryosurgery is reliable as a surgical treatment for enchondroma and ACT. Further research should define the criteria for determining which specific cartilaginous tumors necessitate surgical treatment.

Intralesional treatment versus wide resection for central low-grade chondrosarcoma of the long bones.

BACKGROUND: Grade I or low-grade chondrosarcoma (LGCS) is a primary bone tumour with low malignant potential. Historically, it was treated by wide resection, since accurate pre-operative exclusion of more aggressive cancers can be challenging and under-treatment of a more aggressive cancer could negatively influence oncological outcomes. Intralesional surgery for LGCS has been advocated more often in the literature over the past few years. The potential advantages of less aggressive treatment are better functional outcome and lower complication rates although these need to be weighed against the potential for compromising survival outcomes. OBJECTIVES: To assess the benefits and harms of intralesional treatment by curettage compared to wide resection for central low-grade chondrosarcoma (LGCS) of the long bones. SEARCH METHODS: We searched the Cochrane Central Register of Controlled Trials (CENTRAL; 2018, Issue 4), MEDLINE and Embase up to April 2018. We extended the search to include trials registries, reference lists of relevant articles and review articles. We also searched 'related articles' of included studies suggested by PubMed. SELECTION CRITERIA: In the absence of prospective randomised controlled trials (RCTs), we included retrospective comparative studies and case series that evaluated outcome of treatment of central LGCS of the long bones. The primary outcome was recurrence-free survival after a minimal follow-up of 24 months. Secondary outcomes were upgrading of tumour; functional outcome, as assessed by the Musculoskeletal Tumor Society (MSTS) score; and occurrence of complications. DATA COLLECTION AND ANALYSIS: We used standard methodological procedures recognised by Cochrane. We conducted a systematic literature search using several databases and contacted corresponding authors, appraised the evidence using the ROBINS-I risk of bias tool and GRADE, and performed a meta-analysis. If data extraction was not possible, we included studies in a narrative summary. MAIN RESULTS: We included 18 studies, although we were only able to extract participant data from 14 studies that included a total of 511 participants; 419 participants were managed by intralesional treatment and 92 underwent a wide resection. We were not able to extract participant data from four studies, including 270 participants, and so we included them as a narrative summary only. The evidence was at high risk of performance, detection and reporting bias.Meta-analysis of data from 238 participants across seven studies demonstrated little or no difference in recurrence-free survival after intralesional treatment versus wide resection for central LGCS in the long bones (risk ratio (RR) 0.98; 95% confidence interval (CI) 0.92 to 1.04; very low-certainty evidence). MSTS scores were probably better after intralesional surgery (mean score 93%) versus resection (mean score 78%) with a mean difference of 12.69 (95% CI 2.82 to 22.55; P value < 0.001; 3 studies; 72 participants; low-certainty evidence). Major complications across six studies (203 participants) were lower in cases treated by intralesional treatment (5/125 cases) compared to those treated by wide resection (18/78 cases), with RR 0.23 (95% CI 0.10 to 0.55; low-certainty evidence). In four people (0.5% of total participants) a high-grade (grade 2 or dedifferentiated) tumour was found after a local recurrence. Two participants were treated with second surgery with no evidence of disease at their final follow-up and two participants (0.26% of total participants) died due to disease. Kaplan-Meier analysis of data from 115 individual participants across four studies demonstrated 96% recurrence-free survival after a maximum follow-up of 300 months after resection versus 94% recurrence-free survival after a maximum follow-up of 251 months after intralesional treatment (P value = 0.58; very low-certainty evidence). Local recurrence or metastases were not reported after 41 months in either treatment group. AUTHORS' CONCLUSIONS: Only evidence of low- and very low-certainty was available for this review according to the GRADE system. Included studies were all retrospective in nature and at high risk of selection and attrition bias. Therefore, we could not determine whether wide resection is superior to intralesional treatment in terms of event-free survival and recurrence rates. However, functional outcome and complication rates are probably better after intralesional surgery compared to wide resection, although this is low-certainty evidence, considering the large effect size. Nevertheless, recurrence-free survival was excellent in both groups and a prospective RCT comparing intralesional treatment versus wide resection may be challenging for both practical and ethical reasons. Future research could instead focus on less invasive treatment strategies for these tumours by identifying predictors that help to stratify participants for surgical intervention or close observation.

Radiofrequency ablation in the treatment of atypical cartilaginous tumours in the long bones: lessons learned from our experience.

BACKGROUND: Surgery is the cornerstone of treatment of symptomatic cartilaginous neoplasms. We previously studied the application of radiofrequency ablation of atypical cartilaginous tumours in the long bones. The purpose of the present study was to investigate the additional effect of placing multiple needles and a longer procedure duration on the proportion of completely ablated tumours. Post-ablation MRI findings and the occurrence of complications were also assessed. METHODS: We prospectively included 24 patients with atypical cartilaginous tumours in the long bones. Patients underwent CT-guided radiofrequency ablation followed by curettage with adjuvant phenolisation 3 months later, retrieving material assessed for viable tumour. Before curettage, gadolinium-enhanced MRI was performed to check for residual tumour. The occurrence of complications was noted. RESULTS: Complete tumour ablation was achieved in 17 out of 24 patients (71%). Complete ablation was achieved in 5 of the 6 cases (83%) when multiple needles were used in tumours ≥30 mm. There was incomplete ablation in 8% of patients. Post-ablation gadolinium-enhanced MRI findings agreed with the histological results in 17 out of 23 cases and there was a negative predictive value of 83%. One patient suffered a fracture after radiofrequency ablation. CONCLUSION: Radiofrequency ablation could be an alternative to curettage when treating atypical cartilaginous tumours in the long bones. It was shown that multiple needle placement in addition to longer duration of the ablation procedure is an effective measure in achieving complete ablation in tumours ≥30 mm. Gadolinium-enhanced MRI has a negative predictive value of 83% and could guide post-ablation follow-up.

Local Treatment with Adjuvant Therapy for Central Atypical Cartilaginous Tumors in the Long Bones: Analysis of Outcome and Complications in One Hundred and Eight Patients with a Minimum Follow-up of Two Years.

BACKGROUND: A central atypical cartilaginous tumor (ACT)--formerly known as chondrosarcoma grade 1 (CS1)--is a tumor of intermediate-type malignancy, often treated with surgery. The extent of surgery remains controversial, as some advocate resection and others favor local treatment by curettage. Because of the low prevalence of ACT/CS1, the available data are limited and generally not uniform. The purpose of this study was to present the outcome for a large cohort of patients with ACT/CS1 in the long bones who were treated with curettage and adjuvant phenolization and followed for a minimum of two years according to national guidelines. METHODS: A retrospective study was designed to analyze data from 108 patients treated for central ACT/CS1 in the long bones between 2006 and 2012. All patients were treated with curettage and adjuvant phenolization, and defects were filled with polymethylmethacrylate, bone graft, or bone substitutes. The primary end point was local recurrence or residual tumor. Secondary end points included the type and rate of complications and reoperations. RESULTS: All patients were free from local recurrence at a mean follow-up of 48.7 months (range, 24.3 to 97.5 months). Residual tumor was suspected in five patients, leading to a 95.4% disease-free survival rate. A fracture occurred in eleven patients (10.2%). Other complications were osseous penetration during the surgery (two patients), wound infection (one patient), arthrofibrosis (one patient), and skin necrosis (one patient). Tumor volume was related neither to the risk of fracture nor to the occurrence of residual tumor. CONCLUSIONS: In our experience, curettage of ACT/CS1 in the long bones with adjuvant phenolization is safe, even with large tumors of up to 100 cm(3). Most worrisome is the risk of fracture, which occurred in 10.2% of our patients. Considering the relatively mild behavior of ACT/CS1, less aggressive treatment, by observation or by minimally invasive surgery, could be the next step that should be evaluated prospectively.

Conservative treatment of a pathological fracture in a 3-year-old boy with primary hyperoxaluria type I.

Primary hyperoxaluria type I is a rare inherited disease that presents a disturbed metabolism of glyoxylate. Consequently, patients suffer from hyperoxaluria, leading to renal failure and subsequent skeletal calcium oxalate deposition. Areas with high concentrations of calcium oxalate, so-called dense metaphyseal bands, are at risk for pathological fracturing. The primary disease is treated by combined liver-kidney transplantation, although pathological fracturing also occurs in the posttransplant period. In the current case, we present a 3-year-old boy with a pathological fracture of his right femur, 2 years after liver-kidney transplantation. We opted for a conservative regime, leading to good fracture healing. As there are limited data in the literature regarding treatment of fractures in these patients, it is important to notify the outcome of conservative treatment of pathological fractures in patients with primary hyperoxaluria type I.

Hip fracture after radiofrequency ablation therapy for bone tumors: two case reports.

Radiofrequency ablation (RFA) has become a valuable therapeutic modality in cancer treatment over the last decade. In orthopedic surgery, RFA is used for the treatment of benign bone tumors and bone metastases. Complications are rare and, to our knowledge, bone fracture as a complication due solely to RFA has not been reported to date. In this report we describe two patients with a fracture in the calcar region of the femur as a complication of RFA treatment for bone malignancies. Since RFA is applied increasingly often, it is important to report this risk of fracture as a complication of treatment of lesions in the femoral calcar.