論文 Does re-ossification after palliative radiotherapy for spinal bone metastases help maintain vertebral body height? The spine journal : official journal of the North American Spine Society,23(10):1540-1548 2023(Oct.) Author:Munehisa Kito; Yoshinori Tsukahara; Masanori Okamoto; Ayumu Fukazawa; Shota Ikegami; Atsushi Tanaka; Yukiko Komatsu; Hirokazu Ideta; Kaoru Aoki; Yasunari Fujinaga; Jun Takahashi Abstract:BACKGROUND CONTEXT: After palliative radiotherapy of spinal bone metastases, re-ossification is sometimes observed in bone with osteolytic changes. However, it remains unknown whether the re-ossification that is observed after radiotherapy is associated with preservation of vertebral body height. PURPOSE: To investigate whether re-ossification observed after palliative radiotherapy can contribute to the preservation of vertebral body height. STUDY DESIGN: This is a retrospective observational study. PATIENTS SAMPLE: We investigated 111 vertebral bodies in 54 patients that underwent palliative radiotherapy at a single center for painful osteolytic/mixed metastatic spinal tumors in solid tumors between 2016 and 2020. OUTCOME MEASURES: The outcome measures were the presence of re-ossification and vertebral body height reduction on the CT image. METHODS: Re-ossification was evaluated according to the MD Anderson response classification criteria, and sagittal CT images were used to evaluate vertebral body height. A vertebral body ID was assigned to the irradiated vertebral body, and continuous CT images obtained for each vertebral body ID were evaluated. The median number of evaluation periods for each vertebral body was 4, and the total number of periods was 463. Logistic regression analysis was performed to investigate factors related to the occurrence of vertebral body height reduction before the subsequent CT. As a subanalysis, factors related to re-ossification were investigated. RESULTS: The following primary cancer types were observed: lung cancer, 41 vertebral bodies; breast cancer, 19; renal cell cancer, 15; other, 36. A total of 62.2% showed re-ossification. The median time to confirmation of re-ossification by CT was 2 months. Factors significantly associated with vertebral body height reduction were presence of vertebral body height reduction before radiotherapy (odds ratio [OR] 6.8, 95% confidence interval [CI] 2.0-63, p=.01) and no re-ossification (OR 137, 95% CI 22-3469, p<.01). Factors associated with re-ossification were the type of cancer and total radiation dose. Those with lung cancer and those with a total radiation dose of 20 Gy or less were more prone to re-ossification. CONCLUSIONS: Re-ossification was observed in 62.2% of vertebral bodies after palliative radiotherapy for painful osteolytic/mixed metastatic spinal tumors. The re-ossification group demonstrated significantly less vertebral body height reduction when compared with the non-re-ossification group. The presence of re-ossification may potentially serve an important role in maintaining vertebral body height.
Two-Staged Reconstruction of Extensor Tendons After Wide Resection of Clear Cell Sarcoma at the Anterior Ankle: A Case Report. JBJS case connector,13(3) 2023(Jul. 01) Author:Yukiko Komatsu; Akira Takazawa; Munehisa Kito; Hiroshi Yamazaki; Jun Takahashi; Hiroyuki Kato Abstract:CASE: A 23-year-old woman sustained a skin defect in the anterior ankle involving the extensor retinaculum (ER) and 10 cm of extensor tendons (ETs) after a wide resection of clear cell sarcoma. The skin defect was reconstructed with free latissimus dorsi flap, ER with tensor fascia lata, and ETs with 2-staged tendon reconstruction using silicone rod interposition, followed by semitendinosus and gracilis tendon grafts. Four years postoperatively, she achieved 92% of ankle and 70% of great toe motion of the contralateral side. CONCLUSION: This reconstruction technique was useful for a large soft-tissue defect at the anterior ankle.
Muscle strength and functional recovery for soft-tissue sarcoma of the thigh: a prospective study. International journal of clinical oncology,28(7):922-927 2023(Jul.) Author:Atsushi Tanaka; Masanori Okamoto; Munehisa Kito; Yasuo Yoshimura; Kaoru Aoki; Shuichiro Suzuki; Akira Takazawa; Yukiko Komatsu; Hirokazu Ideta; Takaaki Ishida; Jun Takahashi Abstract:BACKGROUND: This study aimed to investigate changes in muscle strength and functional outcome before and after surgery for soft-tissue sarcoma of the thigh and to examine the timing of recovery. METHODS: From 2014 to 2019, 15 patients who underwent multiple resections of the thigh muscle for soft-tissue sarcoma of the thigh were included in this study. The muscle strength was measured with an isokinetic dynamometer for the knee joint and with a hand-held dynamometer for the hip joint. The functional outcome assessment was based on the Musculoskeletal Tumor Society (MSTS) score, Toronto Extremity Salvage Score (TESS), European Quality of Life-5 Dimensions (EQ-5D), and maximum walking speed (MWS). All measurements were conducted preoperatively and at 3, 6, 12, 18, and 24 months postoperatively, and the ratio of postoperative to preoperative value was used. A repeated-measures analysis of variance was performed to compare changes over time and to investigate the recovery plateau. Correlations between changes in muscle strength and functional outcomes were also examined. RESULTS: The muscle strength of the affected limb, MSTS score, TESS, EQ-5D, and MWS were significantly decreased at 3 months postoperatively. The recovery plateau was subsequently reached at 12 months postoperatively. The changes in muscle strength of the affected limb and functional outcome showed a significant correlation. CONCLUSIONS: The estimated postoperative recovery for soft-tissue sarcoma of the thigh is 12 months after surgery.
What Are the Complications, Function, and Survival of Tumor-devitalized Autografts Used in Patients With Limb-sparing Surgery for Bone and Soft Tissue Tumors? A Japanese Musculoskeletal Oncology Group Multi-institutional Study. Clinical orthopaedics and related research 2023(Jun. 14) Author:Akihiko Takeuchi; Hiroyuki Tsuchiya; Nokitaka Setsu; Tabu Gokita; Yasunori Tome; Naofumi Asano; Yusuke Minami; Hiroyuki Kawashima; Suguru Fukushima; Satoshi Takenaka; Hidetatsu Outani; Tomoki Nakamura; Satoshi Tsukushi; Teruya Kawamoto; Teruki Kidani; Munehisa Kito; Hiroshi Kobayashi; Takeshi Morii; Toru Akiyama; Tomoaki Torigoe; Koji Hiraoka; Akihito Nagano; Shigeki Kakunaga; Kazuhiko Hashimoto; Makoto Emori; Hisaki Aiba; Yoshikazu Tanzawa; Takafumi Ueda; Hirotaka Kawano Abstract:BACKGROUND: Tumor-devitalized autografts treated with deep freezing, pasteurization, and irradiation are biological reconstruction methods after tumor excision for aggressive or malignant bone or soft tissue tumors that involve a major long bone. Tumor-devitalized autografts do not require a bone bank, they carry no risk of viral or bacterial disease transmission, they are associated with a smaller immunologic response, and they have a better shape and size match to the site in which they are implanted. However, they are associated with disadvantages as well; it is not possible to assess margins and tumor necrosis, the devitalized bone is not normal and has limited healing potential, and the biomechanical strength is decreased owing to processing and tumor-related bone loss. Because this technique is not used in many countries, there are few reports on the results of this procedure such as complications, graft survival, and limb function. QUESTIONS/PURPOSES: (1) What was the rate of complications such as fracture, nonunion, infection, or recurrence in a tumor-devitalized autograft treated with deep freezing, pasteurization, and irradiation, and what factors were associated with the complication? (2) What were the 5-year and 10-year grafted bone survival (free from graft bone removal) of the three methods used to devitalize a tumor-containing autograft, and what factors were associated with grafted bone survival? (3) What was the proportion of patients with union of the tumor-devitalized autograft and what factors were associated with union of the graft-host bone junction? (4) What was the limb function after the tumor-devitalized autograft, and what factors were related to favorable limb function? METHODS: This was a retrospective, multicenter, observational study that included data from 26 tertiary sarcoma centers affiliated with the Japanese Musculoskeletal Oncology Group. From January 1993 to December 2018, 494 patients with benign or malignant tumors of the long bones were treated with tumor-devitalized autografts (using deep freezing, pasteurization, or irradiation techniques). Patients who were treated with intercalary or composite (an osteoarticular autograft with a total joint arthroplasty) tumor-devitalized autografts and followed for at least 2 years were considered eligible for inclusion. Accordingly, 7% (37 of 494) of the patients were excluded because they died within 2 years; in 19% (96), an osteoarticular graft was used, and another 10% (51) were lost to follow-up or had incomplete datasets. We did not collect information on those who died or were lost to follow-up. Considering this, 63% of the patients (310 of 494) were included in the analysis. The median follow-up was 92 months (range 24 to 348 months), the median age was 27 years (range 4 to 84), and 48% (148 of 310) were female; freezing was performed for 47% (147) of patients, pasteurization for 29% (89), and irradiation for 24% (74). The primary endpoints of this study were the cumulative incidence rate of complications and the cumulative survival of grafted bone, assessed by the Kaplan-Meier method. We used the classification of complications and graft failures proposed by the International Society of Limb Salvage. Factors relating to complications and grafted autograft removal were analyzed. The secondary endpoints were the proportion of bony union and better limb function, evaluated by the Musculoskeletal Tumor Society score. Factors relating to bony union and limb function were also analyzed. Data were investigated in each center by a record review and transferred to Kanazawa University. RESULTS: The cumulative incidence rate of any complication was 42% at 5 years and 51% at 10 years. The most frequent complications were nonunion in 36 patients and infection in 34 patients. Long resection (≥ 15 cm) was associated with an increased risk of any complication based on the multivariate analyses (RR 1.8 [95% CI 1.3 to 2.5]; p < 0.01). There was no difference in the rate of complications among the three devitalizing methods. The cumulative graft survival rates were 87% at 5 years and 81% at 10 years. After controlling for potential confounding variables including sex, resection length, reconstruction type, procedure type, and chemotherapy, we found that long resection (≥ 15 cm) and composite reconstruction were associated with an increased risk of grafted autograft removal (RR 2.5 [95% CI 1.4 to 4.5]; p < 0.01 and RR 2.3 [95% CI 1.3 to 4.1]; p < 0.01). The pedicle freezing procedure showed better graft survival than the extracorporeal devitalizing procedures (94% versus 85% in 5 years; RR 3.1 [95% CI 1.1 to 9.0]; p = 0.03). No difference was observed in graft survival among the three devitalizing methods. Further, 78% (156 of 200 patients) of patients in the intercalary group and 87% (39 of 45 patients) of those in the composite group achieved primary union within 2 years. Male sex and the use of nonvascularized grafts were associated with an increased risk of nonunion (RR 2.8 [95% CI 1.3 to 6.1]; p < 0.01 and 0.28 [95% CI 0.1 to 1.0]; p = 0.04, respectively) in the intercalary group after controlling for confounding variables, including sex, site, chemotherapy, resection length, graft type, operation time, and fixation type. The median Musculoskeletal Tumor Society score was 83% (range 12% to 100%). After controlling for confounding variables including age, site, resection length, event occurrence, and graft removal, age younger than 40 years (RR 2.0 [95% CI 1.1 to 3.7]; p = 0.03), tibia (RR 6.9 [95% CI 2.7 to 17.5]; p < 0.01), femur (RR 4.8 [95% CI 1.9 to 11.7]; p < 0.01), no event (RR 2.2 [95% CI 1.1 to 4.5]; p = 0.03), and no graft removal (RR 2.9 [95% CI 1.2 to 7.3]; p = 0.03) were associated with an increased limb function. The composite graft was associated with decreased limb function (RR 0.4 [95% CI 0.2 to 0.7]; p < 0.01). CONCLUSION: This multicenter study revealed that frozen, irradiated, and pasteurized tumor-bearing autografts had similar rates of complications and graft survival and all resulted in similar limb function. The recurrence rate was 10%; however, no tumor recurred with the devitalized autograft. The pedicle freezing procedure reduces the osteotomy site, which may contribute to better graft survival. Furthermore, tumor-devitalized autografts had reasonable survival and favorable limb function, which are comparable to findings reported for bone allografts. Overall, tumor-devitalized autografts are a useful option for biological reconstruction and are suitable for osteoblastic tumors or osteolytic tumors without severe loss of mechanical bone strength. Tumor-devitalized autografts could be considered when obtaining allografts is difficult and when a patient is unwilling to have a tumor prosthesis and allograft for various reasons such as cost or socioreligious reasons. LEVEL OF EVIDENCE: Level III, therapeutic study.
Clinical Outcome of Low-Grade Myofibroblastic Sarcoma in Japan: A Multicenter Study from the Japanese Musculoskeletal Oncology Group. Cancers,15(8) 2023(Apr. 15) Author:Munehisa Kito; Keisuke Ae; Masanori Okamoto; Makoto Endo; Kunihiro Ikuta; Akihiko Takeuchi; Naohiro Yasuda; Taketoshi Yasuda; Yoshinori Imura; Takeshi Morii; Kazutaka Kikuta; Teruya Kawamoto; Yutaka Nezu; Ichiro Baba; Shusa Ohshika; Takeshi Uehara; Takafumi Ueda; Jun Takahashi; Hirotaka Kawano Abstract:This retrospective multicenter study aimed to analyze the clinical features and prognosis of 24 patients diagnosed with LGMS between 2002 and 2019 in the Japanese sarcoma network. Twenty-two cases were surgically treated and two cases were treated with radical radiotherapy (RT). The pathological margin was R0 in 14 cases, R1 in 7 cases, and R2 in 1 case. The best overall response in the two patients who underwent radical RT was one complete response and one partial response. Local relapse occurred in 20.8% of patients. Local relapse-free survival (LRFS) was 91.3% at 2 years and 75.4% at 5 years. In univariate analysis, tumors of 5 cm or more were significantly more likely to cause local relapse (p < 0.01). In terms of the treatment of relapsed tumors, surgery was performed in two cases and radical RT was performed in three cases. None of the patients experienced a second local relapse. Disease-specific survival was 100% at 5 years. A wide excision aimed at the microscopically R0 margin is considered the standard treatment for LGMS. However, RT may be a viable option in unresectable cases or in cases where surgery is expected to cause significant functional impairment.
The usefulness of immunohistochemistry for phosphohistone H3 as a prognostic factor in myxoid liposarcoma. Scientific reports,13(1):4733-4733 2023(Mar. 23) Author:Akira Takazawa; Yasuo Yoshimura; Masanori Okamoto; Atsushi Tanaka; Munehisa Kito; Kaoru Aoki; Takeshi Uehara; Jun Takahashi; Hiroyuki Kato; Jun Nakayama Abstract:Myxoid liposarcoma (MLS) is a common subtype of liposarcoma. Although the prognosis is generally good, there are factors known to be associated with poor prognosis. Accurate indices are important to predict prognosis. We aimed to assess the usefulness of immunohistochemistry for phosphohistone H3 (PHH3) as a potential biomarker in comparison with Ki-67 antigen and other prognostic factors. Twenty-five patients with MLS were evaluated. Age, sex, depth of tumor, tumor size, surgical margin, oncological outcome, histological grade, presence of necrosis, proportion of round cell component (RC%), PHH3 index, and Ki-67 index were examined. Prognostic factors of the examination criteria were statistically analyzed, survival rate analyses were performed using the Kaplan-Meier method, and multivariate analysis was performed using Cox proportional-hazard regression analysis. The number of PHH3-positive tumor cells and the PHH3 and Ki-67 indices demonstrated a statistical correlation with the prognosis of MLS in univariate analysis (P < 0.001, P < 0.001, P = 0.01, respectively). PHH3 index and RC% were significant factors in multivariate analysis (P = 0.03, P = 0.02). The immunohistochemistry of PHH3 may be associated with prognosis and could serve as a valid criterion of histological grade in MLS.
Dedifferentiated liposarcoma in the extremity and trunk wall: A multi-institutional study of 132 cases by the Japanese Musculoskeletal Oncology Group (JMOG). European journal of surgical oncology : the journal of the European Society of Surgical Oncology and the British Association of Surgical Oncology,49(2):353-361 2023(Feb.) Author:Takeshi Morii; Ukei Anazawa; Chiaki Sato; Shintaro Iwata; Makoto Nakagawa; Makoto Endo; Tomoki Nakamura; Kunihiro Ikuta; Yoshihiro Nishida; Robert Nakayama; Toru Udaka; Teruya Kawamoto; Munehisa Kito; Kenji Sato; Jungo Imanishi; Toru Akiyama; Hiroshi Kobayashi; Akihito Nagano; Hidetatsu Outani; Shunichi Toki; Toshihiko Nishisho; Keita Sasa; Yoshiyuki Suehara; Hirotaka Kawano; Takafumi Ueda; Hideo Morioka Abstract:BACKGROUND: Dedifferentiated liposarcoma occurs predominantly in the retroperitoneum. Given the paucity of cases, information on the clinical characteristics of this entity in the extremities and trunk wall is quite limited. In particular, the significance of preoperative evaluation and principles of intraoperative management of the different components, i.e., well-differentiated and dedifferentiated areas, are still to be defined. METHODS: Clinical characteristics, treatment outcomes, and risk factors for poor oncological outcomes in cases of dedifferentiated liposarcoma in the extremity or trunk wall were analyzed by a retrospective, multicentric study. RESULTS: A total of 132 patients were included. The mean duration from the initial presentation to dedifferentiation was 101 months in dedifferentiation-type cases. The 5-year local recurrence-free survival, metastasis-free survival, and disease-specific survival rates were 71.6%, 75.7%, and 84.7%, respectively. Among 32 patients with metastasis, 15 presented with extrapulmonary metastasis. A percentage of dedifferentiated area over 87.5%, marginal/intralesional margin, and R1/2 resection in the dedifferentiated area were independent risk factors for local recurrence. Dedifferentiated areas over 36 cm2, French Federation of Cancer Centers Sarcoma Group grade III, and intralesional or marginal resection were independent risk factors for metastasis. A dedifferentiated area over 77 cm2 and lung metastasis were independent risk factors for disease-specific mortality. CONCLUSIONS: The typical clinical characteristics of dedifferentiated liposarcoma in the extremity and trunk wall were reconfirmed in the largest cohort ever. The evaluation of the dedifferentiated area in terms of grade, extension, and pathological margin, together with securing adequate surgical margins, was critical in the management of this entity.
Leiomyosarcoma of the femur arising from an aneurysmal bone cyst treated seven years before: A case report and literature review. Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association,27(6):1359-1362 2022(Nov.) Author:Tetsuhiko Mimura; Yasuo Yoshimura; Kenji Sano; Nobuhito Araki; Masanori Okamoto; Munehisa Kito; Atsushi Tanaka; Shuichiro Suzuki; Akira Takazawa; Kaoru Aoki
Clinical Outcome of Patients with Pelvic and Retroperitoneal Bone and Soft Tissue Sarcoma: A Retrospective Multicenter Study in Japan. Cancers,14(12) 2022(Jun. 20) Author:Toshiyuki Takemori; Teruya Kawamoto; Hitomi Hara; Naomasa Fukase; Shuichi Fujiwara; Ikuo Fujita; Takuya Fujimoto; Masayuki Morishita; Kazumichi Kitayama; Shunsuke Yahiro; Tomohiro Miyamoto; Masanori Saito; Jun Sugaya; Katsuhiro Hayashi; Hiroyuki Kawashima; Tomoaki Torigoe; Tomoki Nakamura; Hiroya Kondo; Toru Wakamatsu; Munenori Watanuki; Munehisa Kito; Satoshi Tsukushi; Akihito Nagano; Hidetatsu Outani; Shunichi Toki; Shunji Nishimura; Hiroshi Kobayashi; Itsuo Watanabe; Yusuke Demizu; Ryohei Sasaki; Takumi Fukumoto; Takahiro Niikura; Ryosuke Kuroda; Toshihiro Akisue Abstract: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.
Influence of the adductor compartment resection on muscle strength and postoperative function in soft-tissue sarcoma of the thigh. Japanese journal of clinical oncology,52(4):370-374 2022(Apr. 06) Author:Atsushi Tanaka; Masanori Okamoto; Munehisa Kito; Yasuo Yoshimura; Kaoru Aoki; Shuichiro Suzuki; Akira Takazawa; Yukiko Komatsu; Takaaki Ishida; Jun Takahashi Abstract:OBJECTIVE: To predict the muscle strength and postoperative function for soft-tissue sarcoma arising from the adductor compartment of the thigh. METHODS: Between 2003 and 2019, 17 cases that underwent resection of the adductor muscle group (adductor longus, adductor magnus, adductor brevis, gracilis and pectineus) for soft-tissue sarcoma in the adductor compartment of the thigh were included. The muscle strength was measured with an isokinetic dynamometer for the knee joint and with a hand-held dynamometer for the hip joint (ratio of affected to unaffected side). The Musculoskeletal Tumor Society score, Toronto Extremity Salvage Score, European Quality of Life-5 Dimensions and maximum walking speed were used to assess postoperative function and examine correlations with muscle strength. RESULTS: In 13 cases that underwent an isolated resection of the adductor compartment, reduced adduction strength correlated with increased number of resected muscles in the adductor muscle group (P < 0.001). Postoperative function was maintained, showing no correlations with adduction strength. In four cases that underwent combined resections of other compartments, a decrease was observed in adduction strength as well as the muscle strength of other resected muscles, in addition to a decline in postoperative function. In the 4 or 5 adductor muscle resection group, the comparison between isolated and combined resection revealed comparable results for adduction strength but a significant decrease in postoperative function for the combined resection group. CONCLUSIONS: Postoperative function can be preserved for isolated adductor compartment resection. Combined resections of multiple muscles in other compartments and most adductor muscles may result in decreased postoperative function.
Long-Term Results of Kyocera Modular Limb Salvage System after Resection of Tumors in the Distal Part of the Femur: Report from Japanese Musculoskeletal Oncology Group Study. Cancers,14(4) 2022(Feb. 10) Author:Tomoki Nakamura; Akihiko Matsumine; Yu Toda; Satoshi Takenaka; Hidetatsu Outani; Tomohiro Fujiwara; Yoshihiro Nishida; Satoshi Tsukushi; Yasunori Tome; Teruya Kawamoto; Munehisa Kito; Naohiro Shinohara; Masato Tomita; Tomoaki Torigoe; Akihiro Sudo; Hirotaka Kawano Abstract:BACKGROUND: The distal femur is a common site of bone tumors. After surgical resection, prosthetic replacement is a major reconstruction method. We aimed to elucidate the long-term outcomes of the Kyocera Modular Limb Salvage (KMLS) systems after resection of tumors in the distal part of the femur. METHODS: Between 1998 and 2014, 125 patients were treated at 14 institutions. There were 59 males and 66 females, with a mean age of 35 years. The mean follow-up period was 132 months. RESULTS: There had been 65 additional surgeries, including 56 revisions and 9 amputations: 15 for aseptic loosening, 14 for stem breakage, 13 for deep infection, 13 for rotator-hinge bushing failure, 5 for local recurrence, and 5 for others. Implant survival rates at 10 and 15 years were 58.5% and 39.4%. The cumulative incidence of 15-year revision for femoral stem breakage was 31.7% in patients with cementless fixation. The 15-year cumulative incidence of revision for aseptic loosening was 19.8% in patients with cement fixation. CONCLUSIONS: KMLS systems represent a reliable system with long-term results. Stem breakage should be considered in patients with cementless and/or smaller femoral stem sizes. Aseptic loosening should be considered in patients with cement systems after 10 years.
Points of consideration when performing surgical procedures for proximal femoral bone metastasis. Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association,27(1):229-234 2022(Jan.) Author:Atsushi Tanaka; Masanori Okamoto; Munehisa Kito; Yasuo Yoshimura; Kaoru Aoki; Shuichiro Suzuki; Akira Takazawa; Jun Takahashi Abstract:BACKGROUND: To assess the points of consideration when performing surgical procedures for proximal femoral bone metastasis. METHODS: Fourty patients who underwent surgery for proximal femoral bone metastasis from 2009 to 2019 were included. Prognostic assessments were performed based on the Katagiri scoring system. The low-risk group underwent endoprosthetic replacement (EPR) following resection, while the high-risk group underwent internal fixation (IF). For the intermediate-risk group, one of the surgical procedures was chosen depending on general and local condition. Survival period, walking ability, and treatment failure were evaluated. RESULTS: Sixteen and 24 patients underwent EPR and IF, respectively. Although walking reacquisition rate was 94% (15 of 16 patients) for EPR and 75% (18 of 24 patients) for IF (p = 0.210), the median time to walking was shorter in IF compared to EPR (EPR, 29 days, interquartile range [IQR] 23-40; IF, 14 days, IQR 9-24; p = 0.014). Neither EPR nor IF resulted in treatment failure for those with survival periods of <1 year. The treatment failure rate for those with survival periods of ≥1 year was significantly different between EPR (9%, 1 of 11 patients) and IF (50%, 5 of 10 patients) (p = 0.038). All five patients of treatment failure for IF were renal cancer with no bone formation in the intermediate-risk group. Of the 13 patients who underwent IF for the intermediate-risk group, treatment failure did not occur in 5 patients with bone formation. CONCLUSIONS: Although EPR is a more reliable treatment than IF, patients with life expectancy of <1 year are sufficiently manageable with IF. For the intermediate-risk group, EPR should be selected for cases without expected bone formation, and IF can be considered for cases with expected bone formation due to the risk of treatment failure for IF at ≥ 1 year after surgery.
Antitumor Effect of Sclerostin against Osteosarcoma Cancers,13(23):6015-6015 2021(Nov. 29) Author:Hirokazu Ideta; Kazushige Yoshida; Masanori Okamoto; Jun Sasaki; Munehisa Kito; Kaoru Aoki; Yasuo Yoshimura; Shuichiro Suzuki; Atsushi Tanaka; Akira Takazawa; Hisao Haniu; Takeshi Uemura; Takashi Takizawa; Atsushi Sobajima; Takayuki Kamanaka; Jun Takahashi; Hiroyuki Kato; Naoto Saito Abstract:Various risk factors and causative genes of osteosarcoma have been reported in the literature; however, its etiology remains largely unknown. Bone formation is a shared phenomenon in all types of osteosarcomas, and sclerostin is an extracellular soluble factor secreted by osteocytes that prevents bone formation by inhibiting the Wnt signaling pathway. We aimed to investigate the antitumor effect of sclerostin against osteosarcoma. Osteosarcoma model mice were prepared by transplantation into the dorsal region of C3H/He and BALB/c-nu/nu mice using osteosarcoma cell lines LM8 (murine) and 143B (human), respectively. Cell proliferations were evaluated by using alamarBlue and scratch assays. The migratory ability of the cells was evaluated using a migration assay. Sclerostin was injected intraperitoneally for 7 days to examine the suppression of tumor size and extension of survival. The administration of sclerostin to osteosarcoma cells significantly inhibited the growth and migratory ability of osteosarcoma cells. Kaplan–Meier curves and survival data demonstrated that sclerostin significantly inhibited tumor growth and improved survival. Sclerostin suppressed the proliferative capacity and migratory ability of osteosarcoma cells. Osteosarcoma model mice inhibited tumor growth and prolonged survival periods by the administration of sclerostin. The effect of existing anticancer drugs such as doxorubicin should be investigated for future clinical applications.
Clinical Outcomes of Patients with Metastatic Solitary Fibrous Tumors: A Japanese Musculoskeletal Oncology Group (JMOG) Multiinstitutional Study ANNALS OF SURGICAL ONCOLOGY,28(7):3893-3901 2021(Jul.) Author:Outani, Hidetatsu; Kobayashi, Eisuke; Wasa, Junji; Saito, Masato; Takenaka, Satoshi; Hayakawa, Keiko; Endo, Makoto; Takeuchi, Akihiko; Kobayashi, Hiroshi; Kito, Munehisa; Morii, Takeshi; Imanishi, Jungo; Ueda, Takafumi Abstract:BACKGROUND: Although the unpredictable malignant behavior of solitary fibrous tumors (SFTs) has been recognized, the clinical features and prognosis of metastatic SFTs have not been well documented due to the extreme rarity of these cases. The aim of this study is to investigate the clinical features, prognostic factors, and optimal management of patients with metastatic SFTs. PATIENTS AND METHODS: Sixty patients with metastatic SFT were retrospectively reviewed. Univariate and multivariate analyses were performed to identify the factors associated with survival. Time to next treatment (TNT) was used to evaluate the effects of various chemotherapy regimens. RESULTS: A total of 34 male and 26 female patients (median age 55 years, range, 23-87 years) were included in the study. The median follow-up period after metastasis was 32 months (range 1-126 months). Tumor location and local recurrence were correlated with late metastasis. The 3- and 5-year overall survival rates were 72.7% and 49.2%, respectively. Primary tumor location, number of metastases, and metastasectomy were significantly associated with survival. Metastasectomy was the only significant variable on multivariate analysis. The TNT was significantly different among the various regimens. CONCLUSIONS: Patients with metastatic SFTs had relatively longer survival periods compared with those with other metastatic soft-tissue sarcomas. Tumor location and number of metastases was associated with survival. Surgical resection of the metastatic lesions offers the best chance of survival, however further studies are warranted to define patients who would benefit from metastasectomy, and the most effective chemotherapeutic regimen for patients with metastatic SFTs remains unknown.
Clinical Outcomes of Patients with Metastatic Solitary Fibrous Tumors: A Japanese Musculoskeletal Oncology Group (JMOG) Multiinstitutional Study ANNALS OF SURGICAL ONCOLOGY,28(7):3893-3901 2021(Jul.) Author:Outani, Hidetatsu; Kobayashi, Eisuke; Wasa, Junji; Saito, Masato; Takenaka, Satoshi; Hayakawa, Keiko; Endo, Makoto; Takeuchi, Akihiko; Kobayashi, Hiroshi; Kito, Munehisa; Morii, Takeshi; Imanishi, Jungo; Ueda, Takafumi;
Distal femoral impaction bone grafting in revision for tumor endoprosthesis. The Knee,29:42-48 2021(Mar.) Author:Munehisa Kito; Masanori Okamoto; Kaoru Aoki; Atsushi Tanaka; Yukiko Komatsu; Shuichiro Suzuki; Akira Takazawa; Yasuo Yoshimura; Jun Takahashi Abstract:BACKGROUND: Reconstruction using tumor endoprosthesis has been widely used in cases with large bone defects caused by bone and soft tissue tumor resection of the distal femur which extend into the knee joint. However, reconstruction failure can lead to major problems in the long term. We have been performing impaction bone grafting with allogeneic cancellous bone during revision surgery for tumor endoprosthesis of the distal femur to compensate for the thinness and fragility of the remaining femur. The aim of this study is to examine the surgical method, problems, and clinical outcomes of revision surgery with impaction bone grafting. METHODS: Three patients who underwent revision surgery for tumor endoprosthesis using impaction bone grafting at our institution with more than 2 years of follow-up were included. RESULTS: Union between the graft and host bone were achieved in all cases. The mean time to radiographic union was 1.0 year (0.6-1.5 years). Although intraoperative penetration to the anterior cortex of the distal femur occurred in 2 cases, there were no postoperative fractures. There were no failures of tumor endoprosthesis at final observation. CONCLUSION: Good short-term results were observed in 3 patients who underwent revision tumor endoprosthesis with impaction bone grafting following a bone and soft tissue tumor resection of the distal femur. The method was considered to be a useful treatment option. Impaction bone grafting is an established surgical option that can be applied to revision surgery for tumor endoprosthesis.
Usefulness of surgical treatment for asymptomatic patients with extra-peritoneal desmoid-type fibromatosis: a systematic review and meta-analysis. Japanese journal of clinical oncology,50(5):574-580 2020(May 05) Author:Munehisa Kito; Akira Ogose; Masahiro Yoshida; Yoshihiro Nishida Abstract:OBJECTIVE: The purpose of this systematic review is to assess and compare the efficacy of surgical treatment for patients with asymptomatic extra-peritoneal desmoid-type fibromatosis to the wait-and-see policy by evaluating (1) the exacerbation rate (exacerbation; recurrence after surgery or progressive disease following non-surgical treatment) and (2) treatment-associated complications in extra-peritoneal desmoid-type fibromatosis. METHODS: We evaluated documents published between 1 January 1990 and 31 August 2017. The risk of bias in the selected literature was analyzed using the Cochrane Collaboration Risk of Bias Tool. Quality of evidence was evaluated using Grading of Recommendation, Assessment, Development and Evaluation approach. RESULTS: One prospective cohort study, four case-control studies and five case series studies were identified. Meta-analysis was performed to evaluate the exacerbation rate after treatment on one prospective cohort study and four case-control studies. In comparing surgical and non-surgical treatments, the exacerbation rate was significantly higher in the surgical treatment group (odds ratio: 1.32, 95% confidence interval 1.01-1.73, P = 0.05). However, in the case series study, the recurrence rate was 23.4% for the surgical treatment group, while the progressive disease rate was 28.1% for the non-surgical treatment group. The postoperative complication rates associated with surgical treatment in the two studies were 20.8 and 17.2%, respectively. CONCLUSIONS: When considering the exacerbation rate, non-surgical treatment might be appropriate for asymptomatic patients with extra-peritoneal desmoid-type fibromatosis. However, if patients with tumor-related symptoms opt for surgery, including those who face difficulties due to the presence of tumors, it is important to fully explain to them the possibility that the recurrence rate and treatment-associated functional failures may increase depending on the site of occurrence.
Ultrastructure of CIC-DUX4 sarcoma: the first pathological report. Ultrastructural pathology,44(2):237-244 2020(Mar. 03) Author:Hisashi Tamada; Mikiko Kobayashi; Kenji Sano; Takeshi Uehara; Yuki Matsumoto; Ayako Tateishi; Maki Ohya; Munehisa Kito; Kaoru Aoki; Hiroyuki Kanno Abstract:CIC-DUX4 sarcoma (CDS) is a recently identified subtype of small round cell sarcoma. Morphologically, CDS partially resembles Ewing sarcoma (ES) and has been classified as "ES-like sarcoma"; however, detailed clinicopathologic and molecular genetic analyses have indicated that CDS is a new independent disease. Many studies have provided light microscopic, immunohistochemical, and genetic information about CDS. However, ultrastructural findings associated with this sarcoma are lacking. The aim of this study was to investigate the ultrastructure of CDS tumors and to compare their features with those of ES. We examined two cytogenetically confirmed CDS cases. We found that, compared to typical ES, CDS presented heterogeneity: in cell density, from tightly packed to loosely unconnected areas; in cell shape, from polygonal to pleomorphic with small processes; and in nuclear shape including round, oval, polygonal, elongated, invaginated, or wrinkled formations. However, abundant glycogen in the cytoplasm and rare cell adhesion apparatus between cells are major similarities between CDS and typical ES. Neuroendocrine granules, which are seen in rare ES cases, could not be identified in these two CDS cases. Although cytogenetic differences can validate a definite diagnosis, ultrastructural features could also provide important information about the differences between CDS and ES.
Continued growth of locally aggressive fibrous dysplasia of 22 years duration after reaching adulthood: a case report. Journal of surgical case reports,2020(2):rjz406 2020(Feb.) Author:Yusuke Yamagishi; Masanori Okamoto; Yasuo Yoshimura; Munehisa Kito; Kaoru Aoki; Jun Takahashi Abstract:Fibrous dysplasia generally stops growing when patients reach adulthood. Locally aggressive fibrous dysplasia is an extremely rare subtype of fibrous dysplasia that is characterized by progressive enlargement after bone maturation, cortical bone destruction and soft tissue invasion but without malignant transformation. At 50 years of age, a tumor was found in the rib of a patient. The tumor gradually enlarged over time and imaging findings suggested a malignant tumor. The case was further complicated by restrictive lung disorder. Biopsies from multiple sites showed no malignant findings, and marginal resection with partial curettage was performed. The final diagnosis was locally aggressive fibrous dysplasia, and the restrictive lung disorder improved postoperatively. The natural history of the disease is also unknown. This is the first report in the literature to describe a case in which a lesion exhibited long-term growth over a period of 22 years after reaching adulthood.
Risk factor for wound complications following wide resection of soft tissue sarcoma in the adductor compartment of the thigh. Japanese journal of clinical oncology,49(10):932-937 2019(Oct. 01) Author:Munehisa Kito; Keisuke Ae; Hirotaka Koyanagi; Tabu Gokita; Hideto Furuoka; Masanori Okamoto; Atsushi Tanaka; Shuichiro Suzuki; Akira Takazawa; Kaoru Aoki; Yasuo Yoshimura Abstract:BACKGROUND: The purpose of this study was to investigate deep-seated soft tissue sarcoma (STS) occurring in the adductor compartment of the thigh that underwent wide resection and to clarify the high-risk group for wound complications. PATIENTS AND METHODS: From 2000 to 2017, we reviewed 104 cases of deep-seated STS occurring in the adductor compartment of the thigh that were treated at four specialized facilities with expertise in sarcoma treatment. RESULTS: Wound complications occurred in 40 cases (38.5%), of which 23 cases (22.1%) were cases with major wound complications (MWC). In univariate analysis, BMI (P < 0.01), maximum tumor diameter (P < 0.01), operation time (P < 0.01), amount of intraoperative bleeding (P < 0.01), and intraoperative intervention to the femoral artery and vein (P < 0.01) were significantly associated with wound complications. In multivariate analysis, the associated parameters were BMI (P < 0.01), maximum tumor diameter (P = 0.02), and intraoperative intervention to the femoral artery and vein (P = 0.01). When limited to cases with MWC, univariate analysis showed that maximum tumor diameter (P < 0.01), diabetes mellitus (P = 0.03), operation time (P < 0.01), amount of intraoperative bleeding (P < 0.01), and intraoperative intervention to the femoral artery and vein (P = 0.02) were significantly associated parameters. In multivariate analysis, maximum tumor diameter (P = 0.02) and amount of intraoperative bleeding (P = 0.04) were associated parameters. CONCLUSIONS: For patients with risk factors for wound complications, control of bleeding are crucial when resecting deep-seated STS in the adductor compartment of the thigh. In cases with large tumors, surgeons should be especially cautious of cases requiring interventions that surround the femoral artery and vein in order to attain an appropriate surgical margin.
Successful treatment of adult pleomorphic rhabdomyosarcoma with bone invasion in the lower leg by chemotherapy and biological reconstruction: A case report. Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association,24(5):930-935 2019(Sep.) Author:Atsushi Tanaka; Yasuo Yoshimura; Takashige Momose; Ken'ichi Isobe; Kaoru Aoki; Munehisa Kito; Masanori Okamoto; Shuichiro Suzuki; Kenji Sano; Hiroyuki Kato
Combined treatment using Mohs' paste and neoadjuvant chemotherapy for giant gluteal soft tissue sarcoma with malignant fungating wound: a case report. Journal of surgical case reports,2019(5):rjz137 2019(May) Author:Kazuaki Nemoto; Masanori Okamoto; Munehisa Kito; Kaoru Aoki; Shuichiro Suzuki; Akira Takazawa; Yasuo Yoshimura; Hiroyuki Kato Abstract:A malignant fungating wound is a cutaneous infiltration of malignant tumor or metastatic lesion that develop into ulceration. Local control is often difficult to obtain, because the Quality of Life of patients can decrease considerably due to bleeding, exudation, odor and pain from the wound. There are few studies in the literature that report the use of Mohs' paste for soft tissue sarcoma with malignant fungating wound. We report a case resulting in good local control for a patient with dedifferentiated liposarcoma with gluteal ulceration by the combined use of Mohs' paste and chemotherapy as a pre-operative adjuvant therapy. Mohs' paste controlled the infection, odor and exudation in approximately 2 weeks, and good visualization of the surgical field was obtained due to tumor volume reduction. We found that Mohs' paste is effective as a neoadjuvant therapy for disintegrated soft tissue sarcoma.
Clinical outcome of osteosarcoma and its correlation with programmed death-ligand 1 and T cell activation markers. OncoTargets and therapy,12:2513-2518 2019 Author:Kazushige Yoshida; Masanori Okamoto; Jun Sasaki; Chika Kuroda; Haruka Ishida; Katsuya Ueda; Satomi Okano; Hirokazu Ideta; Takayuki Kamanaka; Atsushi Sobajima; Takashi Takizawa; Munehisa Kito; Kaoru Aoki; Takeshi Uemura; Hisao Haniu; Hiroyuki Kato; Naoto Saito Abstract:PURPOSE: Although both anti-PD-1 antibody and treatments using anti-PD-L1 antibody are currently in clinical use, their therapeutic effects vary according to cancer type. One of the factors accounting for this variability is the expression level of the immune checkpoint molecule that differs between cancer types; thus, it is important to clarify the relationship between clinical outcomes and immune checkpoint molecules for all types of human cancer. The purpose of this study is to evaluate the clinical outcome of osteosarcoma in relation to PD-L1, PRF, GZMB, and IFNγ expression. METHODS: Using 19 clinical specimens of osteosarcoma, we examined the expression of PD-L1, PRF, GZMB, and IFNγ in relation to their clinical outcomes. RESULTS: PD-L1 expression correlated with early metastatic formation in clinical specimens of osteosarcoma, and the group with highly expressed functional markers for T cells such as PRF and GZMB resulted in a long overall survival time. CONCLUSION: This is the first study to elucidate the clinical outcomes of osteosarcoma in relation to PD-L1, PRF, GZMB, and IFNγ expression. This study provides valuable information regarding the clinical indication and prediction of effect for anti-PD-1 antibody in osteosarcoma.
Using the Barthel Index to Assess Activities of Daily Living after Musculoskeletal Tumour Surgery: A Single-centre Observational Study. Progress in rehabilitation medicine,4:20190010-20190010 2019 Author:Masanori Okamoto; Munehisa Kito; Yasuo Yoshimura; Kaoru Aoki; Shuichiro Suzuki; Atsushi Tanaka; Akira Takazawa; Kazushige Yoshida; Yoshikazu Ido; Takaaki Ishida; Keiko Kawasaki; Hiroyuki Kato Abstract:OBJECTIVE: The objective of the current study was to find the factors affecting the activities of daily living, as evaluated by the Barthel Index, at the end of rehabilitation after musculoskeletal tumour surgery. Further, we evaluated whether the Barthel Index correlates with functional scores that are specific to musculoskeletal tumours at final follow-up. METHODS: The activities of daily living of 190 patients who underwent postoperative rehabilitation after surgery to treat musculoskeletal tumours were evaluated at the end of the program using the Barthel Index. Functional evaluation at the time of final follow-up observation was evaluated using the Musculoskeletal Tumour Society Score and the Toronto Extremity Salvage Score. RESULTS: The post-rehabilitation Barthel Index was significantly lower in elderly patients aged more than 60 years and in those with malignant tumours and tumours larger than 10 cm. Malignancy and large tumour size were risk factors for a low Barthel Index. There was significant correlation between the Musculoskeletal Tumour Society Score/Toronto Extremity Salvage Score at final functional evaluation and the Barthel Index at the end of rehabilitation. CONCLUSION: The Barthel Index is a simple method to assess the activities of daily living and can potentially predict disease-specific health-related quality of life at final functional evaluation after musculoskeletal tumour surgery.
Giant cell tumor of the distal femur: Outcome beyond 20 years of follow-up after curettage with polymethylmethacrylate. Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association,23(6):1051-1055 2018(Nov.) Author:Munehisa Kito; Seiichi Matsumoto; Keisuke Ae; Taisuke Tanizawa; Tabu Gokita; Keiko Hayakawa; Yuki Funauchi; Naoki Yamamoto Abstract:BACKGROUND: Polymethymethacrylate (PMMA) is often used to reconstruct defects after curettage of Giant Cell Tumors (GCT). While GCTs usually originate in the epiphysis, the use of PMMA in distal femoral lesions may induce the risk of degenerative osteoarthritis (OA). We investigated the limb function of patients after curettage with PMMA beyond 20 years of follow-up. METHODS: Patients with more than 20 years of follow-up who underwent curettage with PMMA for distal femoral GCTs were observed. We retrospectively investigated the radiographic assessment of OA and functional assessment of the limb. The Kellgren and Lawrence (KL) grading system was used for radiographic evaluation. RESULTS: Five patients were included in this study. The mean age was 33 years, and the mean period from application of PMMA to final follow-up observation was 28.1 years. Four lesions were primary, and one lesion was recurrent. There were no patients with postoperative recurrence. There were no OA changes in preoperative radiographs. The shortest mean distance from PMMA to the articular cartilage was 4.6 mm on radiographs immediately after surgery. On radiographs at final follow-up observation, the KL grading were as follows: grade 1, 2 patients; grade 2, 1 patient; grade 3, 2 patients. All patients were able to independently ambulate without a crutch, and there was not enough pain to require nonsteroidal anti-inflammatory drugs. The mean flexion of the knee joint was 116°. CONCLUSIONS: Although PMMA used for distal femoral GCTs exhibited OA changes beyond a 20 year follow-up period, there were no cases requiring artificial joints, and the affected limbs demonstrated good function.
The status quo of treatment and clinical outcomes for patients over 80 years of age with high-grade soft tissue sarcoma: report from the soft tissue tumor registry in Japan. Japanese journal of clinical oncology,48(10):900-905 2018(Oct. 01) Author:Masanori Okamoto; Munehisa Kito; Yasuo Yoshimura; Kaoru Aoki; Shuichiro Suzuki; Atsushi Tanaka; Akira Takazawa; Kazushige Yoshida; Hiroyuki Kato Abstract:OBJECTIVE: The purpose of this study is to clarify the status quo of management and clinical outcome of treatments for oldest-old (≥80 years) patients with high-grade soft tissue sarcomas in Japan. METHOD: The present study was conducted using data from the Soft Tissue Tumor Registry in Japan. There were 956 oldest-old patients with soft tissue sarcoma who were registered from 2006 to 2012. Among them, cases with incomplete data, low-grade soft tissue sarcoma and those who underwent treatment at other institutions were excluded from analysis. RESULTS: We examined 451 cases of high-grade soft tissue sarcoma in oldest-old patients. Three-hundred fifty-one cases (77.8%) were surgically managed, while 100 cases were conservatively managed. In patients aged 85 years and older, 73.1% underwent surgical treatment. A significantly higher proportion of patients underwent conservative therapy in oldest-old patients aged 85 years or older (P = 0.036), patients with deep-seated tumors (P = 0.027) and patients with distant metastases at the first visit (P = 0.000). The median follow-up period was 18.9 months (range, 0.2-83.1). Risk factors for overall survival were extracompartmental tumor progression (P = 0.014) and presence of distant metastases at the first visit (P = 0.000). CONCLUSION: We reported the status quo of treatment and clinical outcome for oldest-old patients with high-grade soft tissue sarcoma in Japan. Although surgery is the primary treatment for soft tissue sarcoma, a significantly higher proportion of patients underwent conservative therapy over surgical treatment in patients aged 85 years or older, patients with deep-seated tumors and patients with distant metastases.
Multicentric giant cell tumor of bone: Case series of 4 patients. Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association,22(6):1107-1111 2017(Nov.) Author:Munehisa Kito; Seiichi Matsumoto; Keisuke Ae; Taisuke Tanizawa; Tabu Gokita; Keiko Hayakawa; Yuki Funauchi; Yutaka Takazawa Abstract:BACKGROUND: We aimed to retrospectively investigate patients with multicentric giant cell tumor (MCGCT) who were treated at our hospital and to clarify their clinical features, treatment policy, and follow-up method. METHODS: Four patients with two or more giant cell tumor (GCT) that occurred in the same patient were treated at our institution between 1978 and 2015. These patients were evaluated for the following: frequency, age of onset, number and site of occurrence, time to occurrence of the next lesion, treatment, recurrence, malignant transformation, metastasis, and oncological outcome. RESULTS: The rate of occurrence was 1.7%. The average age was 25.2 (17-44). The total number of lesions was three in two cases and two in two cases. All four cases had only one lesion during the initial visit. The most frequent site of occurrence was the proximal femur, followed by two lesions that occurred in the metaphysis. The interval between confirmation of the initial lesion and occurrence of the second lesion was in average 12.1 years (0.8-27.0). Initial presentations of lesions were treated by en bloc resection in one case and curettage in three cases. Local recurrences occurred in two cases that underwent curettage. The six lesions that occurred after the initial lesion were treated as follows: en bloc resection in four lesions, curettage and radiation therapy in one, and embolization and radiation therapy in one. Pathologically, no lesions presented malignancy. Pulmonary metastasis occurred in one case. The oncological outcome was NED in three cases and AWD in one case. CONCLUSIONS: No lesions were malignant, and by providing the same treatment as solitary GCT, the oncological outcome was good. It is unnecessary to be concerned of its risks and postoperatively conduct long-term searches for focal lesions across the body.
Clinical outcomes of patients 80 years of age and older with soft tissue sarcoma. Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association,22(5):951-957 2017(Sep.) Author:Masanori Okamoto; Yasuo Yoshimura; Kaoru Aoki; Munehisa Kito; Atsushi Tanaka; Shuichiro Suzuki; Akira Takazawa; Ken'ichi Isobe; Hiroyuki Kato Abstract:OBJECTIVES: Although soft tissue sarcoma (STS) is rare, its incidence is increasing among older patients. Few studies have compared the outcomes between conservative and surgical treatments for STS patients aged ≥80 years. We assessed the outcomes of both treatments in this population and the association between older age and surgical outcome. METHODS: We recruited consecutive patients with STS aged ≥80 years treated at our institution between January 2006 and May 2014. We recommended surgical resection for all patients without multiple distant metastases. Overall survival and sarcoma-specific survival were assessed using the Kaplan-Meier method. RESULTS: Of the 39 patients with STS who presented at our institution, 37 were included in this analysis (19 men and 18 women with a median age of 85 [range 80-94] years). Tumors were classified as Stage IB (n = 3), IIA (n = 6), IIB (n = 3) or III (n = 24). Four patients underwent conservative therapy and 33 underwent surgical resection. The most common tumor site was the lower extremity, and the majority of tumors were classified as undifferentiated pleomorphic sarcoma. The follow-up rate was 100%. One-year sarcoma-specific survival rates were 25.0% in the conservative therapy group and 90.9% in the surgical resection group. No associations were found between age ≥85 years and perioperative complications or clinical outcome. CONCLUSIONS: Surgical resection had relatively few complications, given the age group, and improved the prognosis of older patients with STS. Surgical resection of STS with curative intent should be considered in older patients.
Pulmonary metastasis from giant cell tumor of bone: clinical outcome prior to the introduction of molecular target therapy. Japanese journal of clinical oncology,47(6):529-534 2017(Jun. 01) Author:Munehisa Kito; Seiichi Matusmoto; Keisuke Ae; Taisuke Tanizawa; Tabu Gokita; Hiroshi Kobayashi; Keiko Hayakawa; Yuki Funauchi Abstract:OBJECTIVE: We analyzed the risk factors for pulmonary metastasis from giant cell tumor of bone and aimed to discuss their therapeutic strategy and appropriate follow-up period. METHODS: We analyzed 141 patients of giant cell tumor of bone. The variables analyzed included age, gender, primary site, Campanacci grading, surgical treatment on the primary lesion, radiotherapy and local recurrence. RESULTS: Pulmonary metastasis occurred in 12 patients. The risk factors were young age, Campanacci Grade III and local recurrence. Median time from initial surgery to metastasis was 1.3 years (0-3.1 years). Among them, eight patients experienced local recurrence of the primary tumor, and the median time from initial surgery to local recurrence was 0.8 years (0.3-2.9 years). Among seven patients who underwent wide resection, three patients showed local recurrence of the soft tissue. Nine patients underwent metastasectomy for pulmonary metastases. Of three patients who did not undergo metastasectomy, one patient died of uncontrollable metastases, and two patients showed no changes in their metastatic lesions. CONCLUSIONS: Although we found a correlation between local recurrence and pulmonary metastasis, we were still unable to prevent local or metastatic recurrence by wide resection. Local recurrence and metastasis have been found within ~3 years after initial surgery, and routine image examinations of the primary site and chest after initial surgical treatment should be considered for at least 3 years postoperatively.
Clinical outcome of dedifferentiated liposarcoma in the extremities: A retrospective case series of 7 patients. Journal of orthopaedic science : official journal of the Japanese Orthopaedic Association,21(5):673-7 2016(Sep.) Author:Munehisa Kito; Yasuo Yoshimura; Ken'ichi Isobe; Kaoru Aoki; Shuichiro Suzuki; Atsushi Tanaka; Masanori Okamoto; Kenji Sano; Hiroyuki Kato Abstract:BACKGROUND: Wide resection is the generally recommended surgical treatment for dedifferentiated liposarcoma (DDLPS) in the extremities. However, it may be appropriate to distinguish the surgical margin of low-grade atypical lipomatous tumor (ALT)/well-differentiated liposarcoma (WDLPS) area from the high-grade dedifferentiated area, because the low- and high-grade areas can be clearly separated, both radiologically and histologically. This study re-evaluated the details of surgical margin of DDLPS in the extremities, and aimed to investigate the optimal surgical margin and the usefulness of adjuvant therapy for DDLPS in the extremities. METHODS: Seven patients diagnosed with DDLPS in the extremities and treated between 1995 and 2013 were analyzed. The use of adjuvant therapy before and after surgery was assessed, and the surgical margins for the ALT/WDLPS and dedifferentiated areas were re-evaluated by using the specimens resected at surgery. Subsequently, the recurrence rates, metastatic rates, and oncological outcomes were examined. RESULTS: Four and three patients had wide (adequate wide margin, n = 3; inadequate wide margin, n = 1) and marginal margins for the dedifferentiated area, respectively, while three and four patients had wide (adequate wide margin, n = 2; inadequate wide margin, n = 1) and marginal margins for the ALT/WDLPS area, respectively. Postoperative radiotherapy was performed in three patients with an inadequate wide margin or a marginal margin for the dedifferentiated area. No patient had local recurrence. Distant metastases occurred in two patients. These patients died of their disease. The other five patients were disease-free. CONCLUSION: The ALT/WDLPS and dedifferentiated areas in the tumor margin may be better to be considered separately in determining the appropriate resection extent for DDLPS in the extremities. Postoperative radiotherapy may provide good local control for cases with a narrow surgical margin.
Chondroitin sulfate synthase 1 expression is associated with malignant potential of soft tissue sarcomas with myxoid substance. Human pathology,50:15-23 2016(Apr.) Author:Takashige Momose; Yasuo Yoshimura; Satoru Harumiya; Ken'ichi Isobe; Munehisa Kito; Mana Fukushima; Hiroyuki Kato; Jun Nakayama Abstract:The glycosyltransferases chondroitin sulfate synthase 1 (CHSY1) and exostoses-like 3 (EXTL3) specifically function in biosynthesis of the glycans chondroitin sulfate and heparan sulfate, respectively. Although these glycans play important roles in pathogenesis of various tumors, their significance in soft tissue sarcoma remains unknown. Here, we asked whether CHSY1 or EXTL3 expression correlates with malignant potential of soft tissue sarcomas with myxoid substance. To do so, we examined 40 samples representing specific types, including 12 cases of myxoid liposarcoma, 14 of myxofibrosarcoma, 12 of malignant peripheral nerve sheath tumor, and 2 of low-grade fibromyxoid sarcoma. We performed immunohistochemistry with anti-CHSY1 and anti-EXTL3 antibodies and compared enzyme expression levels with tumor histologic grade as assessed by the Fédération Nationale des Centres de Lutte Contre le Cancer classification and with patient 5-year survival rate. CHSY1 and EXTL3 were expressed in 72.5% and 32.5% of all tumors, respectively. Notably, CHSY1 was strongly expressed in myxofibrosarcoma and malignant peripheral nerve sheath tumor compared with other tumors and significantly associated with higher- rather than lower-grade tumors (P < .01). High expression of CHSY1 was also significantly associated with poorer patient outcomes (P = .031) and higher stages assessed by American Joint Committee on Cancer staging system (P = .004). By contrast, EXTL3 expression was not correlated with histologic grade or patient prognosis. We conclude that CHSY1 expression is closely associated with malignant potential of soft tissue sarcomas with myxoid substance.
A recurrent solitary fibrous tumor of the thigh with malignant transformation: A case report. International journal of surgery case reports,21:111-4 2016 Author:Yasuo Yoshimura; Kenji Sano; Ken-Ichi Isobe; Kaoru Aoki; Munehisa Kito; Hiroyuki Kato Abstract:INTRODUCTION: We describe an unusual case of a uniformly high-grade malignant solitary fibrous tumor (SFT) of the thigh with recurrence after wide resection in a 31-year-old man. PRESENTATION OF CASE: Our current case showed a long-term benign course before the operation, although the subcutaneous tumor was larger than 10cm at presentation. The SFT was diagnosed by needle biopsy, and wide resection was performed. Histological findings showed proliferation of capillaries surrounded by masses of spindle-shaped cells without any cytologic atypia, and the percentage of MIB-1-positive nuclei was 2.1%. However, a rapidly enlarging recurrent tumor was observed 11 months after the operation. A second wide resection for the recurrent tumor was performed. Histologically, the tumor cells uniformly displayed significant cytologic atypia and pleomorphism, and had 40-50 mitoses per 10 high-power fields. The proportion of MIB-1-positive nuclei was 48%. Consequently, the tumor was diagnosed as a SFT with malignant transformation. DISCUSSION: The malignant transformation described in past studies showed high-grade areas within benign, low-grade, or intermediate-grade SFTs. Therefore, in contrast to our case, uniformly high-grade malignant histological findings at recurrence were not described. CONCLUSION: Even if a tumor is non-malignant during the clinical course, as confirmed by tissue biopsy, the possibility of tumor progression to high-grade sarcoma at recurrence should be considered, and the treatment strategy should be determined carefully.
Case Report: Asymptomatic Osteosarcoma Case Reports in Clinical Medicine,04(12):369 2015 Author:Reiko Yamaguchi; Seiichi Matsumoto; Keisuke Ae; Taisuke Tanizawa; Tabu Gokita; Keiko Hayakawa; Yuki Funauchi; Munehisa Kito; Hirotada Ishii; Noriko Motoi
Locked Metacarpophalangeal Joint of the Middle Finger Caused by a Lipoma in the Flexor Tenosynovium: A Case Report. JBJS case connector,4(3):e64 2014(Aug. 13) Author:Munehisa Kito; Yasuo Yoshimura; Kaoru Aoki; Shigeharu Uchiyama; Takeshi Uehara; Hiroyuki Kato
Intraosseous neurilemmoma of the proximal ulna. International journal of surgery case reports,5(12):914-8 2014 Author:Munehisa Kito; Yasuo Yoshimura; Ken'ichi Isobe; Kaoru Aoki; Takashige Momose; Hiroyuki Kato Abstract:INTRODUCTION: Neurilemmoma is a benign nerve sheath neoplasm commonly located in the soft tissue. Intraosseous neurilemmoma is rare, constituting less than 1% of primary bone tumors. PRESENTATION OF CASE: A 21 year-old woman was presented with left elbow pain of 1-month duration. Plain radiographs showed a well-defined, lytic and expansile lesion of the proximal ulna. Computed tomography revealed cortical destruction and soft tissue extension. Because the tissue of origin for the tumor was uncertain, an open biopsy was performed. The specimens demonstrated a benign spindle cell tumor suggestive of a neurilemmoma, similar to a soft tissue neurilemmoma. The diagnosis of intraosseous neurilemmoma was established. Marginal excision of the soft tissue component and curettage of the lesion in the bone were performed. After 3.5 years of follow up, there is no clinical or radiographic finding to suggest any recurrence. DISCUSSION: The major site of intraosseous neurilemmoma is the mandible. Occurrence in the long bone is particularly rare. Only two cases of intraosseous neurilemmoma involving the bones around the elbow have been reported to our knowledge; these cases arose in the distal humerus. We describe the first case of intraosseous neurilemmoma of the proximal ulna of the left elbow. The recommended treatment is conservative resection and bone grafting, as malignant change is extremely rare. CONCLUSION: Although very rare, intraosseous neurilemmoma should be taken under consideration in the differential diagnosis of painful, radiographically benign-appearing osseous tumor around the elbow.
Preoperative radiographic and histopathologic evaluation of central chondrosarcoma. Archives of orthopaedic and trauma surgery,133(9):1225-31 2013(Sep.) Author:Yasuo Yoshimura; Ken-ichi Isobe; Hideki Arai; Kaoru Aoki; Munehisa Kito; Hiroyuki Kato Abstract:BACKGROUND: Distinguishing grade 1 chondrosarcoma from grade 2 chondrosarcoma is critical both for planning the surgical procedure and for predicting the outcome. We aimed to review the preoperative radiographic and histologic findings, and to evaluate the reliability of preoperative grading. METHODS: We retrospectively reviewed the medical records of 17 patients diagnosed with central chondrosarcoma at our institution between 1996 and 2011. In these cases, we compared the preoperative and postoperative histologic grades, and evaluated the reliability of the preoperative histologic grading. We also assessed the preoperative radiographic findings obtained using plain radiography, computed tomography (CT), and magnetic resonance imaging (MRI). RESULTS: Preoperative histologic grade was 1 in 12 patients, 2 in 4 patients, and 3 in 1 patient. However, 6 of the 12 cases classified as grade 1 before surgery were re-classified as grade 2 postoperatively. In the radiographic evaluation, grade 1 was suspected by the presence of a ring-and-arc pattern of calcification on plain radiography and CT and entrapped fat and ring-and-arc enhancement on MRI. Grades 2 and 3 were suspected by the absence of calcification and the presence of cortical penetration and endosteal scalloping on plain radiography and CT, as well as soft-tissue mass formation on MRI. CONCLUSION: Although the combination of radiographic interpretation and histologic findings may improve the accuracy of preoperative grading in chondrosarcoma, the establishment of a standard evaluation system with the histologic and radiographic findings and/or the development of new biologic markers are necessary for preoperative discrimination of low-grade chondrosarcoma from high-grade chondrosarcoma.
研究者総覧
