Clinical characteristics and prognosis of patients with incidentally discovered chest wall sarcoma compared with those of symptomatic patients.

OBJECTIVE: Sarcomas of the bone and soft tissues are detected after the onset of pain, detectable mass and related symptoms in the absence of a standardized screening examination. However, primary chest wall sarcomas can be incidentally detected upon chest X-ray or computed tomography. Previous studies of incidental primary chest wall sarcomas lack prognosis and disease-specific clinical data. This study aimed to investigate the prognoses of patients with incidental chest wall sarcomas and compare them with those of symptomatic patients. METHODS: This study included 18 patients diagnosed with primary chest wall sarcoma between 2010 and 2023. Patient information such as age, sex, tumour diameter, tumour location, symptoms, treatment, time to treatment initiation, pathological diagnosis and outcome were retrospectively analysed. RESULTS: Among the 18 patients, the sarcomas were incidentally detected in five by chest X-ray and computed tomography in three and two patients, respectively. The pathological diagnoses of the patients were Ewing sarcoma, Chondrosarcoma grade 1, grade 2, periosteal osteosarcoma and malignant peripheral nerve sheath tumour. The patients had no symptoms at the first visit to our hospital, and no lesions in other organs were detected at the time of the initial examination. At the final follow-up, the patients remained disease-free after radical treatment. The tumour sizes of the five patients were significantly smaller than those of patients with symptoms (P = 0.003). CONCLUSIONS: The incidental detection of chest wall sarcomas and consequent early detection and treatment of tumours improves patient prognosis relative to that of symptomatically diagnosed patients.

Assessment of clinical and radiological characteristics of Japanese patients with SAPHO syndrome.

OBJECTIVE: This study aimed to analyse the radiological characteristics and clinical diversity of Japanese patients with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, a heterogeneous disorder. METHODS: Radiographs and clinical information from 115 Japanese patients (female/male: 81/34, mean age at onset: 48.7 years) diagnosed with SAPHO syndrome between January 2007 and December 2020 were retrospectively reviewed. Additionally, the treatment for SAPHO syndrome was explored. RESULTS: Among the 115 patients, 70 patients had complications, including palmoplantar pustulosis, acne, or psoriasis. Imaging studies included bone scintigraphy, magnetic resonance imaging, computed tomography, and positron emission tomography in 71, 58, 70, and 23 patients, respectively. The most frequent lesions were arthritis and hyperostosis of the sternoclavicular joints in 96 patients; spinal lesions, including sacroiliac arthritis were observed in 85 patients. Peripheral aseptic osteitis was observed in 22 patients, and the tibia was involved in 12. The treatments consisted of analgesics, bisphosphonates, conventional synthetic disease-modifying anti-rheumatic drugs, and biologics (tumour necrosis factor inhibitors and interleukin-23p19 inhibitors) in 85, 15, 23, and 10 patients (8 and 2 patients), respectively. CONCLUSION: Sternoclavicular hyperostosis and pustulosis are frequently observed in patients with SAPHO syndrome. Biological agents were more frequently used in patients with peripheral osteitis and arthritis.

Hip Transposition Can Provide Early Walking Function After Periacetabular Tumor Resection: A Multicenter Study.

BACKGROUND: Reconstruction after periacetabular bone tumor resection involves important tradeoffs; large bone grafts or endoprostheses are reported to offer fair walking function in general but can be technically demanding and carry a high risk of severe complications. Conversely, hip transposition avoids implant-related risks, but stability and functional return may be less consistent. Fewer studies are available on hip transposition, which is also appealing in more resource-constrained environments, and little is known about the time course from surgery to functional return after hip transposition. QUESTIONS/PURPOSES: (1) What is the time course of recovery of walking function after hip transposition, especially in the first 6 months? (2) What factors are associated with a greater likelihood of early functional recovery? (3) Is early (2-month) functional recovery associated with a greater likelihood of walking ability and higher Musculoskeletal Tumor Society (MSTS) scores? METHODS: Between 2009 and 2019, six tertiary care centers in Japan treated 48 patients with internal hemipelvectomy for malignant tumors. During that time, the preferred reconstructive approach was hip transposition, and 92% (44 of 48) of our patients were treated with this procedure. Among them, 86% (38 of 44) had follow-up of at least 6 months, had no local recurrence during that time, and were included in our retrospective study. We chose 6 months as the minimum follow-up duration because the endpoints in this study pertained to early recovery rather than reconstructive durability. Hip transposition involved moving the proximal end of the femur (femoral head, resection end of the trochanteric area, and spacers such as prostheses) upward to the underside of the resected ilium or the lateral side of the sacrum if sacroiliac joint resection was performed. The end of the proximal femur was stabilized to the remaining ilium or sacrum using polyethylene tape, polyethylene terephthalate mesh, an iliotibial tract graft, or an external fixator, according to the surgeon's preference. The median age at surgery was 46 years (range 9 to 76 years), there were 23 women and 15 men, and the median follow-up duration was 17 months (range 6 to 110 months). The postoperative time course of functional recovery was assessed with a record review, the timing of functional milestones was identified (wheelchair, walker, bilateral crutches, single crutch or cane, and walking without an aid), and the MSTS score at the final follow-up was assessed. Additionally, demographic and surgical factors were reviewed, and their association with short-term functional recovery and the final functional outcome was analyzed. RESULTS: Patients started using a walker at median postoperative day (POD) 20 (IQR 14 to 36) and with bilateral crutches at median POD 35 (IQR 20 to 57). At POD 60, which was the approximate median date of discharge, 76% (29 of 38) of patients were able to walk using bilateral crutches (the early recovery group) and 24% (nine of 38) of patients were not able to do so (the delayed recovery group). No baseline factors were different between the two groups. The early recovery group had a higher median MSTS score than the delayed recovery group: 57% (range 17% to 90%) versus 45% (13% to 57%) (p = 0.047). Moreover, more patients acquired better function (a single crutch or cane or more) in the early recovery group, with a median of 5 months (95% CI 4 to 11) than did those in the delayed recovery group (median not reached) (p = 0.0006). The HR was 15.2 (95% CI 2.5 to 93). Forty-two percent (16 of 38) underwent additional surgery for wound management. CONCLUSION: It took patients a fair amount of time to recover walking function after hip transposition, and patients who could not walk on bilateral crutches at POD 60 seemed less likely to regain walking function and were likely to have lower MSTS scores thereafter. Wound-related complications were frequent. This method may be a realistic alternative for younger patients who have the strength for a long rehabilitation period or those who want to minimize prosthesis-related complications. Future studies with more patients are necessary to understand the risk factors associated with delayed recovery.Level of Evidence Level III, therapeutic study.

Perioperative Adriamycin plus ifosfamide vs. gemcitabine plus docetaxel for high-risk soft tissue sarcomas: randomised, phase II/III study JCOG1306.

BACKGROUND: This randomised phase II/III trial aimed to determine whether perioperative chemotherapy with gemcitabine plus docetaxel (GD) is non-inferior to the standard Adriamycin plus ifosfamide (AI) in terms of overall survival (OS) in patients with soft tissue sarcoma (STS). METHODS: Patients with localised high-risk STS in the extremities or trunk were randomised to receive AI or GD. The treatments were repeated for three preoperative and two postoperative courses. The primary endpoint was OS. RESULTS: Among 143 enrolled patients who received AI (70 patients) compared to GD (73 patients), the estimated 3-year OS was 91.4% for AI and 79.2% for GD (hazard ratio 2.55, 95% confidence interval: 0.80-8.14, P = 0.78), exceeding the prespecified non-inferiority margin in the second interim analysis. The estimated 3-year progression-free survival was 79.1% for AI and 59.1% for GD. The most common Grade 3-4 adverse events in the preoperative period were neutropenia (88.4%), anaemia (49.3%), and febrile neutropenia (36.2%) for AI and neutropenia (79.5%) and febrile neutropenia (17.8%) for GD. CONCLUSIONS: Although GD had relatively mild toxicity, the regimen-as administered in this study-should not be considered a standard treatment of perioperative chemotherapy for high-risk STS in the extremities and trunk. CLINICAL TRIAL REGISTRATION: jRCTs031180003.

Clinical Outcome of Patients with Pelvic and Retroperitoneal Bone and Soft Tissue Sarcoma: A Retrospective Multicenter Study in Japan.

This study aimed to retrospectively analyze the clinical outcomes of patients with pelvic and retroperitoneal bone and soft tissue sarcoma (BSTS). Overall, 187 patients with BSTS in the pelvis and retroperitoneal region treated at 19 specialized sarcoma centers in Japan were included. The prognostic factors related to overall survival (OS), local control (LC), and progression-free survival (PFS) were evaluated. The 3-year OS and LC rates in the 187 patients were 71.7% and 79.1%, respectively. The 3-year PFS in 166 patients without any distant metastases at the time of primary tumor diagnosis was 48.6%. Osteosarcoma showed significantly worse OS and PFS than other sarcomas of the pelvis and retroperitoneum. In the univariate analyses, larger primary tumor size, soft tissue tumor, distant metastasis at the time of primary tumor diagnosis, P2 location, chemotherapy, and osteosarcoma were poor prognostic factors correlated with OS. Larger primary tumor size, higher age, soft tissue tumor, chemotherapy, and osteosarcoma were poor prognostic factors correlated with PFS in patients without any metastasis at the initial presentation. Larger primary tumor size was the only poor prognostic factor correlation with LC. This study has clarified the epidemiology and prognosis of patients with pelvic and retroperitoneal BSTS in Japan.

Clinical outcome of porous hydroxyapatite/collagen graft on bone defects following curettage of bone tumors.

Hydroxyapatite/collagen (HAp/col) has been reported to be a highly useful bone-like nanocomposite. This study included 33 human patients to investigate the details of the clinical outcomes, which are (1) onset of timing of bone regeneration, (2) replacement by regenerated bone of HAp/col and (3) complications, in human cases grafting HAp/col in large bone defects, following curettage of bone tumors. Porous HAp/col initiated bone regeneration approximately 59 days following the surgery. In 15 cases (45%), complete replacement by newly formed bone was observed 12 months after surgery. On the other hand, incomplete replacement of HAp/col at the final follow-up was observed in 13 cases (39%). In these cases, HAp/col could not be detected in the transparent area of postoperative plain radiographs owing to quick absorption; moreover, it was difficult to distinguish whether the transparent area in plain radiographs was remaining HAp/col, recurrence, or remaining tumor. In addition, larger HAp/col implantation volume (≧15 cm3 ) was associated with poorer result of complete replacement (log-rank, p = .005). Further studies are warranted for the construction of a new artificial bone graft substitute that is more quickly and surely regenerated by newly formed bone in large bone defects.

Radiation Eye Dose for Physicians in CT Fluoroscopy-Guided Biopsy.

It is important to evaluate the radiation eye dose (3 mm dose equivalent, Hp (3)) received by physicians during computed tomography fluoroscopy (CTF)-guided biopsy, as physicians are close to the source of scattered radiation. In this study, we measured the radiation eye dose in Hp (3) received by one physician during CTF in a timeframe of 18 months using a direct eye dosimeter, the DOSIRISTM. The physician placed eye dosimeters above and under their lead (Pb) eyeglasses. We recorded the occupational radiation dose received using a neck dosimeter, gathered CT dose-related parameters (e.g., CT-fluoroscopic acquisition number, CT-fluoroscopic time, and CT-fluoroscopic mAs), and performed a total of 95 procedures during CTF-guided biopsies. We also estimated the eye dose (Hp (3)) received using neck personal dosimeters and CT dose-related parameters. The physician eye doses (right and left side) received in terms of Hp (3) without the use of Pb eyeglasses for 18 months were 2.25 and 2.06 mSv, respectively. The protective effect of the Pb eyeglasses (0.5 mm Pb) on the right and left sides during CTF procedures was 27.8 and 37.5%, respectively. This study proved the existence of significant correlations between the eye and neck dose measurement (right and left sides, R2 = 0.82 and R2 = 0.55, respectively) in physicians. In addition, we found significant correlations between CT-related parameters, such as CT-fluoroscopy mAs, and radiation eye doses (right and left sides, R2 = 0.50 and R2 = 0.52, respectively). The eye dose of Hp (3) received in CTF was underestimated when evaluated using neck dosimeters. Therefore, we suggest that the physician involved in CTF use a direct eye dosimeter such as the DOSIRIS for the accurate evaluation of their eye lens dose.

Standardization of evaluation method and prognostic significance of histological response to preoperative chemotherapy in high-grade non-round cell soft tissue sarcomas.

BACKGROUND: Preoperative chemotherapy is widely applied to high-grade localized soft tissue sarcomas (STSs); however, the prognostic significance of histological response to chemotherapy remains controversial. This study aimed to standardize evaluation method of histological response to chemotherapy with high agreement score among pathologists, and to establish a cut-off value closely related to prognosis. METHODS: Using data and specimens from the patients who had registered in the Japan Clinical Oncology Group study, JCOG0304, a phase II trial evaluating the efficacy of perioperative chemotherapy with doxorubicin (DOX) and ifosfamide (IFO), we evaluated histological response to preoperative chemotherapy at the central review board. RESULTS: A total of 64 patients were eligible for this study. The percentage of viable tumor area ranged from 0.1% to 97.0%, with median value of 35.7%. Regarding concordance proportion between pathologists, the weighted kappa coefficient (κ) score in all patients was 0.71, indicating that the established evaluation method achieved substantial agreement score. When the cut-off value of the percentage of the residual tumor area was set as 25%, the p-value for the difference in overall survival showed the minimum value. Hazard ratio of the non-responder with percentage of the residual tumor < 25%, to the responder was 4.029 (95% confidence interval 0.893-18.188, p = 0.070). CONCLUSION: The standardized evaluation method of pathological response to preoperative chemotherapy showed a substantial agreement in the weighted κ score. The evaluation method established here was useful for estimating of the prognosis in STS patients who were administered perioperative chemotherapy with DOX and IFO. TRIAL REGISTRATION: UMIN Clinical Trials Registry C000000096. Registered 30 August, 2005 (retrospectively registered).

Optimal bone biopsy route to the proximal femur evaluated by computed tomography-based finite element modeling.

INTRODUCTION: The proximal femur (PF) is one of the most common locations of benign cystic lesions. A fracture after bone biopsy is a rare but severe complication. However, the risk of fracture after biopsy of this lesion has not been well-studied. Computed tomography (CT)-based finite element (FE) modeling estimates the elastic modulus and compressive strength enables fracture prediction. This study investigated strength of PF after biopsy by CT-FE modeling and determined the optimum biopsy level and size. MATERIALS AND METHODS: Six male bone tumor patients' (15-38 years) total femur CT data (slice thickness, 0.8-1.0 mm) of the healthy side were obtained. Three different cylindrical bone defect (BD) diameters (10, 15, and 20 mm) were set on the lateral surface of PF at the following levels: level 1, insertion of the gluteus minimums; level 2, lower end of the greater trochanter (GT); level 3, origin of the vastus lateralis; level 4, center of the lesser trochanter (LT); and level 5, lower end of LT using Mechanical Finder software (version 8.0). Virtual loads were applied with incremental increases of 100 N until fracture occurred and the fracture load (FL) was evaluated. RESULTS: For BD with a diameter of 15 and 20 mm, there was a significant difference in the decrease of the mean FL, with an average of 22% at level 4 and 5, and 33%-44% at levels 3 to 5, respectively. At level 1 and 2, no significant decrease in the mean FL was observed regardless of the diameter of BD. CONCLUSION: Biopsies at level 1 and 2 showed no significant decrease in bone strength. However, biopsy at level 1 may contaminate the GT bursas. Therefore, biopsy at level 2 (lower end of GT) can avoid contamination and minimize the effect on bone strength.

Erratum: lncRNA HAR1B has potential to be a predictive marker for pazopanib therapy in patients with sarcoma.

[This corrects the article DOI: 10.3892/ol.2021.12716.].

Occupational Radiation Dose to Eye Lenses in CT-Guided Interventions Using MDCT-Fluoroscopy.

In computed tomography (CT)-guided interventions (CTIs), physicians are close to a source of scattered radiation. The physician and staff are at high risk of radiation-induced injury (cataracts). Thus, dose-reducing measures for physicians are important. However, few previous reports have examined radiation doses to physicians in CTIs. This study evaluated the radiation dose to the physician and medical staff using multi detector (MD)CT-fluoroscopy, and attempted to understand radiation-protection and -reduction methods. The procedures were performed using an interventional radiology (IVR)-CT system. We measured the occupational radiation dose (physician and nurse) using a personal dosimeter in real-time, gathered CT-related parameters (fluoroscopy time, mAs, CT dose index (CTDI), and dose length product (DLP)), and performed consecutive 232 procedures in CT-guided biopsy. Physician doses (eye lens, neck, and hand; µSv, average ± SD) in our CTIs were 39.1 ± 36.3, 23.1 ± 23.7, and 28.6 ± 31.0, respectively. Nurse doses (neck and chest) were lower (2.3 ± 5.0 and 2.4 ± 4.4, respectively) than the physician doses. There were significant correlations between the physician doses (eye and neck) and related factors, such as CT-fluoroscopy mAs (eye dose: r = 0.90 and neck dose: r = 0.83). We need to understand the importance of reducing/optimizing the dose to the physician and medical staff in CTIs. Our study suggests that physician and staff doses were not significant when the procedures were performed with the appropriate radiation protection and low-dose techniques.

lncRNA HAR1B has potential to be a predictive marker for pazopanib therapy in patients with sarcoma.

Bone and soft-tissue sarcomas are rare and are highly heterogeneous mesenchymal malignancies. It is therefore challenging to acquire the clinical data of patients with specific histological subtypes of sarcoma using large clinical trials, and there is a need to further establish the diagnosis and treatment of sarcomas. The results of the current study revealed that long non-coding RNA (lncRNA) highly accelerated region 1B (HAR1B) may serve as a predictive biomarker for pazopanib treatment in bone and soft-tissue sarcomas. Using multiplex reverse transcription-quantitative PCR and microarray analyses, the results demonstrated that HAR1B and HOX transcript antisense RNA (HOTAIR) were differentially expressed in pazopanib-sensitive cells and responders. It was further revealed that small interfering RNA-knockdown of HAR1B led to an increased resistance to pazopanib in sarcoma cell lines. Gene expression profiles associated with pazopanib sensitivity included cellular molecular pathways, such as genes involved in von-Willebrand factor-related signaling. The current study demonstrated that lncRNA HAR1B expression in sarcoma cell lines affected cellular sensitivity to pazopanib in patients with sarcoma.

Bladder perforation by orthopedic implants 26 years after limb-sparing surgery for left proximal femoral chondrosarcoma: A case report.

INTRODUCTION: The need for implant use during orthopedic surgeries has been increasing. Accordingly, increased implant failures have been reported. However, bladder perforation remains a rare complication after orthopedic surgery. Although a few reports have described bladder perforation after total hip arthroplasty, no previous studies have reported the migration of staples into the bladder after limb-sparing surgery. PRESENTATION OF CASE: A 65-year-old patient underwent limb-sparing surgery to remove a chondrosarcoma in the left proximal thigh. Twenty-six years after surgery, a staple that had been used to fix artificial ligaments to the pubis migrated to perforate the bladder, resulting in painful urination. The staple was removed, and her symptoms improved. DISCUSSION: In this case, bladder perforation by the staple resulted in painful urination. The bladder perforation was not detected until 26 years after the initial surgery. CONCLUSION: Our observations emphasize that implant complications may occur even after a long postoperative period, and the possibility of delayed bladder perforation from previous pelvic surgeries should be considered in patients presenting with urinary tract symptoms.

Ten-year follow-up results of perioperative chemotherapy with doxorubicin and ifosfamide for high-grade soft-tissue sarcoma of the extremities: Japan Clinical Oncology Group study JCOG0304.

BACKGROUND: Soft-tissue sarcomas (STS) are rare malignant tumors those are resistant to chemotherapy. We have previously reported the 3-year follow-up result on the efficacy of perioperative chemotherapy with doxorubicin (DXR) and ifosfamide (IFM) for high-risk STS of the extremities (JCOG0304). In the present study, we analyzed the 10-year follow-up results of JCOG0304. METHODS: Patients with operable, high-risk STS (T2bN0M0, AJCC 6th edition) of the extremities were treated with 3 courses of preoperative and 2 courses of postoperative chemotherapy, which consisted of 60 mg/m2 of DXR plus 10 g/m2 of IFM over a 3-week interval. The primary study endpoint was progression-free survival (PFS) estimated by Kaplan-Meier methods. Prognostic factors were evaluated by univariable and multivariable Cox proportional hazards model. RESULTS: A total of 72 patients were enrolled between March 2004 and September 2008, with 70 of these patients being eligible. The median follow-up period was 10.0 years for all eligible patients. Local recurrence and distant metastasis were observed in 5 and 19 patients, respectively. The 10-year PFS was 65.7% (95% CI: 53.4-75.5%) with no PFS events being detected during the last 5 years of follow-up. The 10-year overall survival was 78.1% (95% CI: 66.3-86.2%). Secondary malignancy was detected in 6 patients. The subgroup analysis demonstrated that there was significant difference in survival with regard to primary tumor size. CONCLUSIONS: Only a few long-term results of clinical trials for perioperative chemotherapy treatment of STS have been reported. Our results demonstrate that the 10-year outcome of JCOG0304 for patients with operable, high-risk STS of the extremities was stable and remained favorable during the last 5 years of follow-up. TRIAL REGISTRATION: This trial was registered at the UMIN Clinical Trials Registry as C000000096 on August 30, 2005.

Arthroscopic Excision of Intra-Articular Osteoid Osteoma at the Elbow.

Osteoid osteoma (OO) apparent in the intra-articular region of the elbow is very rare. Although computed tomography-guided excision and radiofrequency ablation have been recognized as useful treatment options, arthroscopic excision has recently received focus as an alternative strategy for lesions close to neurovascular structures or intra- and juxta-articular lesions. We herein report a 17-year-old female who underwent arthroscopic treatment for intra-articular OO located at the olecranon/coronoid fossa. Her symptoms included elbow pain that was exacerbated at night and contracture of elbow flexion-extension, and she was diagnosed with intra-articular OO after 12 months of symptomatic history. Arthroscopically, thorough synovectomy for both the anterior and posterior aspects of the joint enabled definition of the tumor margin with hyperemic alteration and excision of the lesion as an en bloc specimen. At the 12-month follow-up, the patient had no recurrence of elbow limitation or pain. This case report describes the advantages of arthroscopic treatment, including a low-invasive approach and easy accessibility to the whole intra-articular space, which can provide clear visualization of the tumorous lesion.

Osteofibrous dysplasia arising in the humerus: A case report.

Osteofibrous dysplasia is a benign fibro-osseous lesion of bone which is most commonly occurred in cortical bone of anterior mid-shaft of the tibia of infancy and childhood. This study reported a case of osteofibrous dysplasia arising in the humerus of adult, resulting in good prognosis after a surgical treatment. A 34-year-old male had felt left upper arm pain and was suspected as having a bone tumor at the humeral shaft by X-ray pictures. The tumor was suspected as the osteofibrous dysplasia of the humerus by a core needle biopsy. Intralesional curettage, intraoperative anhydrous ethanol therapy, and artificial bone graft were performed. Surgical specimens showed fibro-osseous lesion, which strongly indicated osteofibrous dysplasia. Seven years after the surgery, he has lived without any local recurrence and complaints. It is important to recognize that osteofibrous dysplasia can arise in the humerus of an older patient for appropriate diagnosis.

Clinical outcome of primary giant cell tumor of bone after curettage with or without perioperative denosumab in Japan: from a questionnaire for JCOG 1610 study.

BACKGROUND: Giant cell tumor of bone (GCTB) is an intermediate tumor known to be locally aggressive, but rarely metastasizing. To plan a prospective study of GCTB, we performed a questionnaire survey for institutions participating in the Bone and Soft Tissue Tumor Study Group (BSTTSG) in the Japan Clinical Oncology Group (JCOG) in 2015. METHODS: We reviewed 158 consecutive patients with primary GCTB treated with curettage without perioperative denosumab from 2008 to 2010 in Japan. We investigated local and distant recurrence rates after definitive curettage. We also investigated the recurrence rate after treatment with preoperative and/or postoperative denosumab with curettage in recent years. There were 40 patients treated with perioperative denosumab, and the factors affecting recurrence in them were investigated. RESULTS: Answers were available from 24 of 30 institutions (80.0%) participating in JCOG BSTTSG. Thirty (19.0%) and 4 (2.5%) of 158 patients developed local and distant recurrence after curettage without perioperative denosumab from 2008 to 2010, respectively. Campanacci grade and embolization before surgery were significantly associated with increasing incidence of local recurrence after curettage (p = 0.034 and p = 0.022, respectively). In patients treated with perioperative desnosumab, 120 mg denosumab was administered subcutaneously for a median 6 (2-41) and 6 (1-14) times in preoperative and postoperative settings, respectively. The recurrence rates were 6 of 21 (28.6%), 2 of 9 (22.2%), and 0 of 10 (0.0%) in the preoperative, postoperative, and both pre- and postoperative denosumab treatment groups, respectively. With all of the preoperative treatments, administration exceeding five times was significantly associated with a decreased incidence of local recurrence after curettage (p < 0.001). CONCLUSION: The recurrence rate of GCTB was still high after curettage, especially in Campanacci grade III, and improvements in the therapeutic strategy are needed in this cohort. There is a possibility that a sufficient dose of preoperative denosumab can reduce recurrence after curettage. Recently, we have started a clinical trial, JCOG1610, to investigate the efficacy of preoperative denosumab in patients who can be treated with curettage in GCTB.

Clinical features and radiological findings of 67 patients with SAPHO syndrome.

OBJECTIVES: The purpose of this study was to facilitate the understanding of the SAPHO (Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis) syndrome by analyzing the clinical and radiological features of 67 Japanese patients with SAPHO syndrome. METHODS: Sixty-seven Japanese patients (female/male: 44/23, mean age at onset: 48.5 years) were diagnosed with SAPHO syndrome from 2002 to 2013 at our hospital. Medical records and radiological imaging of these patients were retrospectively reviewed. RESULTS: Among the 67 patients, 41 had dermatological manifestations, such as palmoplantar pustulosis, acne, and psoriasis. Initial symptom was local pain in all patients, and the most common initial site of the symptom was the anterior chest. Bacterial and fungal cultures from 20 bone biopsies were all negative. Histopathological diagnosis of the specimens was non-specific inflammation in all cases. Bone lesions were observed in 65 patients (97.0%). On the other hand, articular lesions including enthesitis were found in 31 patients (46.2%). CONCLUSION: SAPHO syndrome had different clinical and radiological aspects. The clinical features were not remarkable, except the dermatological manifestations and the involvement of the anterior chest. Bone lesions including hyperostosis and osteitis were found radiographically in the majority of patients with SAPHO syndrome. These are the characteristics of the SAPHO syndrome, with the exclusion of other bone diseases.

Factors associated with the decision of operative procedure for proximal femoral bone metastasis: Questionnaire survey to institutions participating the Bone and Soft Tissue Tumor Study Group of the Japan Clinical Oncology Group.

BACKGROUND: Pathological fracture of the proximal femur is a main cause of cancer patients losing their ability to walk. Although both osteosynthetic devices (predominantly intramedullary nails) and prosthetic replacement have been widely performed for treatment, controversies exist regarding which procedure should be used for the various conditions. In order to decide the eligibility criteria of a planned randomized prospective study about the treatment of pathological fractures of the proximal femur, we assessed the factors affecting the selection of operative procedures using questionnaires sent to the members of the Bone and Soft Tissue Tumor Study Group (BSTTSG) of the Japan Clinical Oncology Group (JCOG). METHODS: Questionnaire surveys to evaluate (1) the priority levels of the factors, (2) the equipoise range of each factor in situations where either procedure could be applied, (3) risk and benefit of each procedure, and (4) the degree of bone destruction affecting the selection of operative procedures, were sent to 26 institutions. RESULTS: Over 80% of the institutions answered. Orthopaedic surgeons of BSTTSG decided on the procedure according to the following factors in descending order: life expectancy, performance status before fracture, the degree of bone destruction, walking ability before fracture, general complications, the number of bone metastases in other sites, and the visceral metastasis status. With regard to bone destruction, (1) the involvement of the head, neck, calcar, and intertrochanteric region, (2) transverse destruction >1/2, and (3) soft-tissue tumor extension, were the factors that led to the choice of prosthesis treatment. CONCLUSIONS: Using these identified factors, the inclusion criteria for the prospective randomized study of the surgical treatment of metastatic bone tumors of the proximal femur were optimized. The evaluation system about the bone destruction of metastases needs to be refined through the following prospective randomized study.