A Natural Organic Compound "Decursin" Has Both Antitumor and Renal Protective Effects: Treatment for Osteosarcoma.

Osteosarcoma is a rare malignant tumor that commonly occurs in children. Anticancer drugs, for example, cisplatin, aid in postsurgery recovery but induce side effects such as renal damage, affecting the life prognosis of patients. Decursin which is one of the bioactive components has been reported for its anti-inflammatory, antioxidant, and antitumor effects, but the effect on osteosarcoma is unexplained. In this study, the research theme was to examine the sensitizing effect of decursin and its influence on cisplatin-induced nephrotoxicity. The cell viability and half maximal inhibitory concentration (IC50), apoptosis induction, and effect on cell cycle and Akt pathways were examined. In vivo, we examine the effects of decursin on tumors and mice bodies. Additionally, the effects of the cisplatin-decursin combination were evaluated in vitro and in vivo. Decursin suppressed cell viability and induced apoptosis via the cell cycle. Decursin also inhibited the Akt pathway by suppressing the phosphorylation of Akt. It enhanced apoptosis induction and lowered cell viability in combination with cisplatin. The increasing tumor volume was suppressed in the decursin-administrated group with further suppression in combination with cisplatin compared to sole cisplatin administration. The decrease in renal function and renal epithelial cell damage caused by cisplatin was improved by the combinatorial treatment with decursin. Therefore, decursin demonstrated an antitumor effect on the osteosarcoma cells and a renal protective effect in combination with cisplatin. Therefore, decursin is a prospective therapeutic agent against osteosarcoma.

Pleomorphic liposarcoma of the extremity with solitary huge liver metastasis at initial diagnosis treated with conversion surgery combined with adjuvant chemotherapy: a case report.

BACKGROUND: Pleomorphic liposarcoma is the rarest subtype of liposarcoma. Pleomorphic liposarcomas are generally unresponsive to chemotherapy and radiotherapy. Moreover, metastasis in the liver, as the first and sole site, from a primary extremity soft tissue sarcoma, including pleomorphic liposarcoma, is extremely rare. Information regarding the appropriate management of these lesions is limited. CASE PRESENTATION: A 50-year-old Japanese woman presented with a mass in the left thigh. Imaging examination revealed a soft tissue sarcoma on the left posterior thigh. The tumor was histologically diagnosed as pleomorphic liposarcoma. Computed tomography examination for assessment of metastases incidentally detected a huge liver mass. Wide excision of sarcoma was performed prior to chemotherapy. Right trisectionectomy was necessary to achieve hepatic clearance; however, the future liver remnant volume was insufficient. Therefore, we decided to administer anthracycline-based chemotheraphy to shrink the tumor. After seven courses of adriamycin-based chemotherapy, the liver tumor size was reduced from 211 mm × 106 mm × 180 mm to 105 mm × 66 mm × 90 mm. Finally, a right hemihepatectomy was performed. The patient was continuously monitored and was metastasis or local recurrence free within 5 months after liver surgery. CONCLUSION: Chemotherapy is effective in some cases for the treatment of unresectable liver metastases of pleomorphic liposarcoma, and complete resection is possible with conversion surgery. If the patient's general condition permits, anthracycline-based chemotherapy can be used for the treatment of stage 4 pleomorphic liposarcoma.

Pristimerin inhibits the proliferation of HT1080 fibrosarcoma cells by inducing apoptosis.

Fibrosarcoma is a soft tissue sarcoma that is classified as a rare cancer. Therefore, no standard anti-tumor drug therapy has been established for fibrosarcoma. Although pristimerin (PM) has been reported to exert an anti-tumor effect on various types of cancer, no studies have examined the therapeutic effect of PM on soft tissue sarcoma. The purpose of the current study was to investigate the anti-tumor effect of PM on human fibrosarcoma cells (HT1080). The present study examined the cell viability, IC50 values and ability to induce apoptosis of PM in HT1080 and normal human dermal fibroblast (aHDF) cells. The effect of PM on the following signaling pathways associated with cell proliferation was also evaluated: AKT and mitogen-activated protein kinase (MAPK). Using mice subcutaneously transplanted with fibrosarcoma cells, the effect of PM treatment was investigated on tumor growth inhibition, body weight and liver and renal function. The results revealed that PM administration reduced cell viability and induced apoptosis in a dose-dependent matter. In HT1080 cells, the IC50 value of PM was 0.16 µM at 24 h and 0.13 µM at 48 h. PM treatment also decreased the levels of phosphorylated AKT, mTOR, NF-κB and phosphorylated ERK in a dose-dependent manner. In the PM injection group, the increase in tumor volume was significantly reduced and the effect on weight loss and liver and renal function were revealed to be insignificant. PM exerted little effect on normal human dermal fibroblasts and was highly effective against human fibrosarcoma cells. The results indicated that PM may be used as a potential therapeutic agent against fibrosarcoma.

A retrospective study of antibacterial iodine-coated implants for postoperative infection.

Postoperative infection is one of the most serious complications in orthopedic surgery. We have developed and use iodine-coated implants to prevent and treat postoperative infection in compromised hosts. This study evaluated outcomes using iodine-coated implants for postoperative infections.We treated 72 postoperative infected patients using iodine-coated implants. Of these, 38 were males and 34 were females, with a mean age of 59.3 years. The mean follow-up period was 5.6 years. The patients included 23 with an infection following total knee arthroplasty, 20 following total hip arthroplasty, 11 following osteosynthesis, 11 following spine surgery, 6 following tumor excision, and 1 following osteotomy. Of these, 37 underwent single-stage surgery and 35 underwent staged revision surgery. We performed staged surgery in any case with active infection. The survival of iodine-coated implants was determined using Kaplan-Meier analysis. White blood cell (WBC) and C-reactive protein (CRP) levels were measured pre- and postoperatively. To evaluate the systemic effects of iodine, serum thyroid hormone levels were examined.Five patients underwent re-revision surgery. In 3 patients, periprosthetic infection recurred at an average of 18 months after surgery. The reinfection rate was 4.2%. These patients recovered following reimplantation of iodine-coated prostheses. No patients required amputation. The survival rate of iodine-coated implants was 91%. There were no signs of infection at the latest follow-up. The median WBC level was nearly in the normal range, and CRP levels returned to normal within 4 weeks after surgery. No abnormalities of thyroid gland function were detected.Iodine-coated titanium implants can be very effective in the treatment of postoperative infections. An iodine coating can be safely applied to infected regions.

The tetraspanin CD81 mediates the growth and metastases of human osteosarcoma.

PURPOSE: CD81 is a member of the tetraspanin family of membrane proteins. Recently, it has been shown that CD81 may be involved in cancer cell proliferation and metastasis. As yet, however, there have been few reports on the expression and role of CD81 in osteosarcoma. METHODS: The expression of CD81 was investigated in human osteoblast cell line hFOB1.19 and in human osteosarcoma cell lines Saos2, MG63 and 143B. The expression of CD81 was inhibited in osteosarcoma cells using siRNA after which cell proliferation, migration and invasion were assessed. We also used Western blotting to investigate the phosphorylation status of Akt, Erk, JNK and p38, and measured the expression of MMP-2, MMP-9 and MT1-MMP. In addition, we used a CRISPR/Cas9 system to stably knock out CD81 expression in 143B cells, transplanted the cells into mice, and assessed tumor formation and lung metastasis in these mice compared to those in the control group. RESULTS: We found that CD81 was expressed in the human osteoblast cell line and in all osteosarcoma cell lines tested. The osteosarcoma cell line 143B exhibited a particularly high level of expression. In addition, we found that osteosarcoma cell proliferation, migration and invasion were decreased after CD81 inhibition, and that the phosphorylation of Akt and Erk was suppressed. Also, the expression levels of MMP-2, MMP-9 and MT1-MMP were found to be suppressed, with MMP-9 showing the greatest suppression. In vivo, we found that mice transplanted with CD81 knockout 143B cells exhibited significantly less tumor formation and lung metastasis than mice in the control group. CONCLUSION: Based on our findings we conclude that inhibition of CD81 suppresses intracellular signaling and reduces tumorigenesis and lung metastasis in osteosarcoma cells.

Chondroid lipoma of the neck: a case report.

BACKGROUND: Chondroid lipoma, first described in 1993 by Meis and Enzinger, is a very rare lipomatous tumor. Because it is a benign tumor, it does not require radiotherapy, chemotherapy, or extensive resection. However, histologically, it is often confused with a sarcoma. It is crucial to differentiate chondroid lipoma from sarcoma to avoid choosing an inappropriate treatment strategy. Although MRI, radiography, and ultrasound have been used to evaluate chondroid lipomas, imaging cannot accurately differentiate chondroid lipoma from sarcoma. CASE PRESENTATION: A 39-year-old man presented to a local clinic with a 1-month history of a painless mass in his left neck. Results of a needle biopsy suggested an atypical lipomatous tumor, and the patient was referred to our hospital. Physical examination revealed a hard and mobile mass in the left neck. Plain X-ray radiographs showed an absence of calcification in the soft tissue mass. MRI revealed a well-defined and lobulated mass, and on T1-weighted images, the lesion showed heterogeneity, with higher signal intensity than that of muscle. On T2-weighted images, the septum had low-signal intensity. On T2-weighted fat-suppressed images, the signal of the mass was completely suppressed. The SUVmax of the mass on FDG PET was 1.84. An additional needle biopsy was performed, and on the basis of the results, we arrived at a diagnosis of well-differentiated liposarcoma. The mass was resected marginally. Macroscopically, the mass was encapsulated and markedly harder than well-differentiated liposarcoma. Histologically, the tumor was composed of myxoid and cartilaginous matrix, and mature fat cells and lipoblast-like cells were present. The final diagnosis was chondroid lipoma, and no recurrence was observed 1 year after surgery. CONCLUSIONS: Chondroid lipoma is an extremely rare benign soft tissue tumor that is often confused with sarcoma. It is very important to differentiate chondroid lipoma from sarcoma when the SUVmax value of the mass is low, even when biopsy results suggest that it is a sarcoma.

Hepatic metastases from primary extremity leiomyosarcomas: Two case reports.

INTRODUCTION: Leiomyosarcoma is a highly malignant soft tissue sarcoma. Most leiomyosarcomas of the extremities metastasize initially to the lungs, with few metastasizing to the liver. Also, it is difficult to diagnose metastases to other regions of the lung during follow-up. CASE PRESENTATION: The first patient was a 51-year-old Japanese woman diagnosed with a leiomyosarcoma of the left distal femur. She underwent chemotherapy, followed by wide tumor excision and reconstruction using frozen autograft with total knee arthroplasty. Eleven months later, a focal lesion was observed in her right liver, despite the absence of lung metastases. Partial hepatic resection was performed, and the hepatic lesion was diagnosed a metastasis of leiomyosarcoma. Two years later, there has been no evidence of local recurrence. The second patient was a 60-year-old Japanese male diagnosed with a leiomyosarcoma of the left thigh. He underwent preoperative chemotherapy followed by wide excision. Three years later, a focal lesion was found in his medial liver, despite the absence of lung metastases. Partial hepatic resection was performed, and the hepatic lesion was diagnosed as a metastasis of leiomyosarcoma. At the latest follow-up, there has been no evidence of local recurrence. CONCLUSIONS: The lung is the most common site of metastases from leiomyosarcomas of the extremeties, because these metastases are hematogenous. Both our patients presented with metastases of the liver, despite the absence of lung metastases. Hepatic metastasis is commonly found in computed tomography (CT) scan. Periodic CT scans of the chest and abdomen are necessary in following-up patients who undergo resection of primary leiomyosarcomas of the extremities.

Limb saving surgery for Ewing's sarcoma of the distal tibia: a case report.

BACKGROUND: Ewing's sarcoma is a primary malignant tumor of bone occurring mostly in childhood. Few effective reconstruction techniques are available after wide resection of Ewing's sarcoma at the distal end of the tibia. Reconstruction after wide resection is especially difficult in children, as it is necessary to consider the growth and activity of the lower limbs. CASE PRESENTATION: A 12-year-old Japanese boy had presented with right lower leg pain at age 8 years. Imaging examination showed a bone tumor accompanied by a large extra-skeletal mass in the distal part of his tibia. The tumor was histologically diagnosed as Ewing's sarcoma. The patient received chemotherapy, followed by wide resection. Reconstruction consisted of a bone transport method involving external fixation of Taylor Spatial Frame. To prevent infection after surgery, the external fixation pin was coated with iodine. One year after surgery, the patient showed poor consolidation of bone, so iliac bone transplantation was performed on the extended bones and docking site of the distal tibia. After 20 months, tibia formation was good. Three years after surgery, there was no evidence of tumor recurrence or metastases; bone fusion was good, and he was able to run. CONCLUSIONS: The bone transport method is an effective surgical method of reconstruction after wide resection of a bone tumor at the distal end of the tibia, if a pin can be inserted into the distal bone fragment. Coating external fixation pins with iodine may prevent postoperative infection.

A novel arthroscopic all-inside suture technique using the Fast-Fix 360 system for repairing horizontal meniscal tears in young athletes: 3 case reports.

RATIONALE: Considering the risk of osteoarthritis following resection of a horizontally torn meniscus of the knee, repairing and preserving the meniscus as much as possible is preferred. We report 3 cases of restoration of horizontally torn menisci using a novel arthroscopic method we have called "all-inside interleaf vertical suture" that afforded preservation. PATIENT CONCERNS: The 3 patients (aged 14, 17, and 21 years) had knee pain through sports activity. DIAGNOSES: All patients had horizontal tears in the posteromedial part of the meniscus. INTERVENTIONS: The method uses Fast-Fix, whereby a first anchor is inserted from the tibial surface of the tear's superior leaflet and a second anchor is inserted from the femoral surface of the tear's inferior leaflet, and the 2 leaflets are closed using vertical suture. In all cases, the suture knots were embedded between the superior leaflet and inferior leaflet, avoiding contact with the articular cartilage, and superior leaflet and inferior leaflet crimping was good. OUTCOMES: All 3 were able to resume competing in sport and ≥ 1 year after surgery they had no pain and their postoperative mean Lysholm scores were 99.7. There were no complications or recurrence. On magnetic resonance imaging, the signal intensity of all the horizontal tears was high before surgery but low after surgery, suggesting that the repaired tear was healing. LESSONS: The all-inside interleaf vertical suture procedure is a new surgical technique that can repair posteromedial horizontal meniscal tears of the knee of young people by easy crimping of the superior and inferior leaflets without the suture knots causing complications.

Antitumor effects of pristimerin on human osteosarcoma cells in vitro and in vivo.

There are very few treatments for musculoskeletal tumors, compared to other cancers; thus, novel therapeutic drugs are needed. Pristimerin (PM) is a triterpene compound isolated from plant extracts that reportedly has antitumor effects on various cancers, such as of the breast and prostate. The purpose of this study was to evaluate the antitumor effects of PM on human osteosarcoma cells. Treatment of the human osteosarcoma cell lines, MNNG and 143B, with PM led to a dose-dependent decrease in cell viability. The effects of PM on apoptosis were evaluated with the Annexin V/propidium iodide assay and analysis of caspases 3, 8, and 9 activities. Western blot analysis showed that PM caused a decrease in the expression of Akt, mTOR, and NF-κB. The volumes and weights of human osteosarcoma xenografts decreased significantly with PM treatment. The results of this study revealed that PM can inhibit human osteosarcoma growth in vitro and in vivo, and may be a novel therapeutic agent for the disease.

Suppression of heat shock protein 70 by siRNA enhances the antitumor effects of cisplatin in cultured human osteosarcoma cells.

Although advances in chemotherapy have improved the prognosis for osteosarcoma, some patients do not respond sufficiently to treatment. Heat shock protein 70 (Hsp70) is expressed at high levels in cancer cells and attenuates the therapeutic efficacy of anticancer agents, resulting in a poorer prognosis. This study investigated whether small interfering RNA (siRNA)-mediated inhibition of Hsp70 expression in an osteosarcoma cell line would enhance sensitivity to cisplatin. The expression of Hsp70 with cisplatin treatment was observed by using Western blotting and real-time reverse transcription polymerase chain reaction (RT-PCR). Changes in the IC50 of cisplatin when Hsp70 was inhibited by siRNA were evaluated. Cisplatin's effectiveness in inducing apoptosis was assessed by assay of terminal deoxynucleotidyl transferase-mediated dUTP nick-end labeling (TUNEL), caspase-3 activity, and mitochondrial membrane potential. Up-regulation of Hsp70 expression was dependent on the concentration of cisplatin. Inhibition of Hsp70 expression significantly reduced the IC50 of cisplatin. When cisplatin was added to osteosarcoma cells with Hsp70 expression inhibited, a significant increase in apoptosis was demonstrated in TUNEL, caspase-3, and mitochondrial membrane potential assays. Inhibition of Hsp70 expression induced apoptosis in cultured osteosarcoma cells, indicating that Hsp70 inhibition enhanced sensitivity to cisplatin. Inhibition of Hsp70 expression may provide a new adjuvant therapy for osteosarcoma.

Subperiosteal inflammatory pseudotumor mimicking primary malignant bone tumor: A case report.

We report a case of IgG4-positive inflammatory pseudotumor mimicking malignant bone tumor. Biopsy revealed no tumor cells. Surgical excision was performed and an abscess developing beneath the periosteum was observed with Streptococcus constellatus. Preoperative serum IgG4 value of 120 mg/dl normalized postoperatively to 80.6 mg/dl. It was difficult to distinguish inflammatory pseudotumor from sarcoma because it developed under the periosteum. In such cases, it is important to measure blood IgG4 values and perform tissue staining and culturing.

The outcomes of reconstruction using frozen autograft combined with iodine-coated implants for malignant bone tumors: compared with non-coated implants.

OBJECTIVE: We perform reconstruction using frozen tumor bone treated by liquid nitrogen after excision of malignant bone tumors. To prevent post-operative infection, we use iodine-coated implants that we developed. The purpose of this study is to compare the outcome of reconstruction using frozen autograft with non-coated implants (group N) and iodine-coated implants (group I). METHODS: Sixty-two patients were included in group N. The mean age was 31.9 ± 2.3 years. A total of 20 patients died and two were lost to follow-up, averaging 20.0 ± 2.9 months post-operatively, leaving 40 patients available for an assessment at a mean of 79.1 ± 5.8 months post-operatively. There were 38 patients in group I. The mean age was 29.8 ± 3.9 years. The mean follow-up period was 32.1 ± 3.0 months. All patients were alive at the latest follow-up. Survival of frozen bone was determined by Kaplan-Meier analysis. RESULTS: In group N, survival of frozen bone was 80.7 ± 6.0% and 57.4 ± 10.2% at 5 and 10 years, respectively. Complications were encountered in 31 of 62 patients (50.0%), including deep infection in 10 (16.1%), fracture in 11 (17.7%), local soft-tissue recurrence in 6 (9.7%) and bone absorption in 4 (6.5%). In group I, survival of frozen bone was 86.7 ± 6.3% at 5 years. Complications were encountered in 8 of 38 patients (21.1%), including deep infection in one (2.6%), fracture in four (10.5%), local soft-tissue recurrence in two (5.3%) and bone absorption in one (2.6%). There was a significantly lower infection rate in group I (P = 0.032). CONCLUSION: Reconstruction using frozen autograft combined with iodine-coated implants for patients with malignant bone tumor is very useful method in which good limb function can be gained with minimized risk of infection.

Metastasis of differentiated thyroid cancer in the subchondral bone of the femoral head: a case report.

BACKGROUND: Differentiated thyroid cancer (DTC) is relatively rare and can metastasize to both the lungs and bones. The great majority of bone metastases occur in red marrow regions where blood flow is high. Only one patient has been described with direct DTC metastasis to the subchondral bone of the femoral head. CASE PRESENTATION: The patient was a 68-year-old Japanese female who had presented with left hip joint pain at age 63 years. At age 51 years, she had been diagnosed with DTC and underwent partial excision. X-rays showed partial femoral head collapse, suggesting osteoarthritis or idiopathic necrosis of the left femoral head. Three years later, a (131) I whole-body scan showed accumulation in the left femoral head, resulting in a diagnosis of DTC metastasis to the left femoral head. Bipolar hip arthroplasty was performed. Examination of the excised femoral head resulted in a final diagnosis of metastasis of follicular thyroid cancer, which was limited histopathologically to the subchondral bone of the femoral head. CONCLUSION: Tumor metastasis to the subchondral bone of the femoral head is exceedingly rare. Overall survival of patients with bone metastasis is improved by complete resection. Differential diagnosis of patients with a previous history of DTC who present with femoral head collapse should include bone metastasis of DTC.

Treatment of a Simple Bone Cyst Using a Cannulated Hydroxyapatite Pin.

Simple bone cysts (SBCs) are benign bone tumors. However, the treatment of SBCs remains controversial because of their healing rate and the invasiveness of surgery. The purpose of the present study was to evaluate the treatment of SBCs using a cannulated hydroxyapatite (HA) pin.A total of 43 patients (35 males, 8 females; mean age 12.1 years; age range, 5-22 years) with SBCs were treated with continuous decompression by inserting ceramic HA pins between 1989 and 2014. The SBCs were located in the calcaneus in 23, the humerus in 15, the femur in 3, and the pelvis in 2 cases. In all patients, minimal fenestration of the cyst wall and curettage and multiple drilling in the cyst wall were performed, followed by insertion of the HA pin. The mean follow-up period was 26.6 months. Operating time, healing period, risk factors for recurrence, and the cure rate were evaluated.Healing was achieved without intervention in 38 patients after a mean of 6.4 months. Two patients had persistent small residual cysts, which had no changes after 1 year at the latest follow-up. There were 5 patients with recurrences (humerus 4, femur 1), who were cured by curettage and artificial bone grafting. The final healing rate by cannulation only using an HA pin was 88.2%. On Fisher exact test, age, site of SBCs, and distance from the physis were found to be significantly associated with SBC recurrence (P < 0.05).In the present study, cannulation using an HA pin for SBCs was found to be a useful technique, particularly for calcaneal cysts, because it is a minimally invasive procedure with a high cure rate. In patients <10 years, involvement of the humerus and contact with the growth plate were significant risk factors for SBC recurrence.

Intramuscular spindle cell lipoma of the deltoid: a case report.

INTRODUCTION: Spindle cell lipoma is an uncommon adipocytic tumor. Intramuscular lesions of this tumor are very rare. In this report, we describe a case of a patient with intramuscular spindle cell lipoma localized in a deltoid. CASE PRESENTATION: A 58-year-old Japanese man visited us because of a soft tissue mass on the lateral aspect of the left shoulder that had been noticed 2 years prior. The spherical tumor, which measured 5 cm × 4 cm, was elastic and firm on palpation and immobile. Magnetic resonance imaging revealed that the lesion was localized in the left deltoid muscle. A needle biopsy was performed to make a histological diagnosis. With a pre-operative diagnosis of intramuscular lipoma, we removed the tumor with the patient under general anesthesia. The tumor was removed with surrounding musculature and fascia. The pathological diagnosis was intramuscular spindle cell lipoma in the left deltoid muscle. CONCLUSIONS: There are several kinds of lipomas. Spindle cell lipoma is a relatively rare variant (1.5% of all adipocytic neoplasms) that is histologically distinct and characterized by the replacement of mature fat by a mixture of mature adipocytes and undifferentiated spindle cells. There are only five other reported cases of intramuscular spindle cell lipoma in the literature, to our knowledge. The case of our patient is very interesting, as to date there have been few reported patients with a diagnosis of an intramuscular spindle cell lipoma in a deltoid.

Primary pulmonary synovial sarcoma requiring differentiation from pulmonary metastasis of tibial adamantinoma: a case report.

BACKGROUND: Primary pulmonary synovial sarcoma (PPSS) is rare. We describe a case of PPSS complicated by tibial adamantinoma that required differentiation from lung metastasis. CASE PRESENTATION: A 39-year-old Japanese woman presented with hemoptysis, dyspnea, and a well-defined tumor measuring 3.0 cm in greatest diameter in the right lower lobe on chest computed tomography (CT). Positron emission tomography/CT with fluorodeoxyglucose (FDG-PET/CT) showed mild uptake of FDG (maximum standardized uptake value of 2.0). Her past history included surgery for adamantinoma of the right tibia at age 25 years. We considered the possibility of pulmonary metastasis from the adamantinoma and performed fluoroscopy-assisted thoracoscopic resection of the tumor after CT-guided Lipiodol marking. Histologically, the tumor was composed mainly of a dense proliferation of spindle cells. Immunohistochemical studies were positive for epithelial membrane antigen, B cell lymphoma 2, and transducing-like enhancer of split 1. They were negative for CD34. The synovial sarcoma, X breakpoint 1 gene-fusion transcript was detected by reverse transcription-polymerase chain reaction. It is diagnostic of PPSS. Resection margins were negative. The patient was well without evidence of recurrence or metastasis of the PPSS or adamantinoma at the 30-month and 15-year follow-ups. CONCLUSION: Clinical and radiological manifestations of PPSS overlap with those of other lung tumors. The solitary pulmonary nodule in this case was indistinguishable from pulmonary metastases of the adamantinoma based on clinical symptoms, epidemiology, chest radiography, CT, and FDG-PET/CT. PPSS was diagnosed only after evaluating gross pathology, histology, immunohistochemistry, and cytogenetics. PPSS should be included in the differential diagnosis of a well-defined homogeneous round or oval lung mass. To our knowledge, this is the first report of PPSS complicated by adamantinoma.