Recurrent lumbar-origin osteoblastoma treated with multiple surgery and carbon ion radiotherapy: a case report.

BACKGROUND: Although osteoblastoma is an uncommon benign bone tumor, it sometimes behaves in a locally aggressive fashion. We herein report a case of recurrent lumbar spine osteoblastoma that was treated by repeated surgery and carbon ion radiotherapy. CASE PRESENTATION: A 13-year-old Japanese girl presented with left side lumbar pain. Computed tomography and magnetic resonance imaging of the lumbar spine demonstrated a tumorous lesion in the left side pedicle of L4. Although gross total resection of the mass, including the nidus, was performed in the initial surgery, recurrence was observed repeatedly in the short term and the pathological diagnosis of all of the resected tumors was conventional osteoblastoma. We finally performed carbon ion radiotherapy after the patient's 3rd palliative operation, and achieved a good outcome. No further recurrence has been observed in 10 years of follow-up. CONCLUSION: We performed carbon ion radiotherapy for a case of recurrent spinal osteoblastoma and achieved a good outcome without recurrence at 10 years after carbon ion radiotherapy treatment. To the best of our knowledge, this is the first case of osteoblastoma that was treated with carbon ion radiotherapy after multiple surgeries.

ß2-Adrenergic receptor expression is associated with biomarkers of tumor immunity and predicts poor prognosis in estrogen receptor-negative breast cancer.

PURPOSE: Antitumor immunity plays an important role in the progression of breast cancer. ß2-adrenergic receptor (ß2AR) was found to regulate the antitumor immune response and breast cancer progression in preclinical studies. To understand the clinical role of ß2AR in cancer progression, we investigated the clinicopathological and prognostic significance of ß2AR expression in invasive breast cancer. METHODS: ß2AR levels in breast tumors were evaluated by immunohistochemistry in a well-characterized patient cohort with long-term follow-up (n = 278). We evaluated the relationship of ß2AR expression to patient survival and clinicopathological factors, including immune biomarkers such as tumor-infiltrating lymphocytes (TILs) and programmed death ligand 1 (PD-L1) expression. Breast cancer-specific survival was compared between high- and low-ß2AR expression groups. RESULTS: Although ß2AR was not related to clinicopathological factors across the whole cohort, high ß2AR was significantly related to PD-L1 negativity in estrogen receptor (ER)-negative patients. Tumors with high ß2AR tended to have low TIL grade, and high ß2AR was an independent prognostic factor for reduced survival in ER-negative patients. CONCLUSIONS: ß2AR is an independent poor prognostic factor in ER-negative breast cancer. The findings suggest that tumor ß2AR regulates immune checkpoint activity, which may have therapeutic implications for patients with ER-negative breast cancer.

Histopathological changes in tumor budding between biopsy and resected specimens from patients treated with preoperative S-1 chemotherapy for oral cancer.

BACKGROUND: S-1 is an oral anticancer agent containing tegafur, a prodrug of 5-fluorouracil (5-FU). Preoperative S-1 chemotherapy is performed to prevent tumor proliferation before surgery in oral squamous cell carcinoma (OSCC). The aim of this study was to evaluate the effects of preoperative S-1 chemotherapy on tumor budding. METHODS: We examined 248 cases of OSCC and evaluated the budding scores in biopsy and resected specimens by keratin immunohistochemistry. RESULTS: In S-1-untreated patients, the budding scores in resected specimens tended to show a mild increase. S-1 treatment had the effect of lowering the budding score, although some cases with dramatically increased budding scores in resected specimens were found in the early phase of administration. Relapse-free survival showed a significant difference (P < .01) according to the presence or absence of S-1 treatment among patients with high budding scores. CONCLUSIONS: The present findings indicate that S-1 administration may be more effective in patients with high budding scores than in those with lower budding scores.

L-type amino acid transporter-1 and CD98 expression in bone and soft tissue tumors.

L-type amino acid transporter-1 (LAT1) is expressed in many cancers. We examined LAT1 and CD98 expression immunohistochemically in surgically resected specimens of various bone and soft tissue tumors. Out of 226 cases, 79 (35%) were LAT1(+) and 95 (42%) were CD98(+) . In bone tumors, LAT1 was highly expressed in osteoblastoma (89%), chondrosarcoma (50%), and osteosarcoma (60%); in soft tissue tumors, LAT1 was highly expressed in rhabdomyosarcoma (80%), synovial sarcoma (63%), Ewing's sarcoma (60%), epithelioid sarcoma (100%) and angiosarcoma (100%). In malignant soft tissue tumors, LAT1 expression was associated with higher histological grade. High CD98 expression was seen in many bone tumors of intermediate and high malignancy. Among soft tissue tumors, CD98 was expressed in tendon sheath giant cell tumor and malignant peripheral nerve sheath tumor (57%), Ewing's sarcoma (50%) and undifferentiated sarcoma (64%). Some of the malignant soft tissue tumors expressed both LAT1 and CD98. This study showed that LAT1 and CD98 was expressed in many malignant and intermediate bone tumors, and some malignant soft tissue tumors.

Solitary osteochondroma of the trapezium: case report.

Osteochondroma of the carpal is rare. We found only 1 case of osteochondroma of the trapezium in the literature. We present a case of a 52-year-old woman with an osteochondroma of the left trapezium and trapeziometacarpal arthritis.

Pathological and molecular analysis of a composite lymphoma of mantle cell lymphoma and Epstein-Barr virus-positive follicular lymphoma.

Composite lymphoma (CL) is a very rare clinical entity defined by the presence of two or more different subtypes of lymphoma in the same lymph node. We report a case of CL in a 78-year-old male presenting with leukocytosis and swelling of multiple lymph nodes. A left axillary node biopsy showed atypical lymphocytes in both the interfollicular and follicular areas. Immunohistochemistry revealed that mantle cell lymphoma (MCL) was mainly present in the interfollicular area and follicular lymphoma (FL) was present in the follicular area. Polymerase chain reaction analysis of immunoglobulin heavy chain gene rearrangements confirmed that they were clonally related neoplasms. However, Epstein-Barr virus (EBV) DNA was detected in only FL cells, suggesting that MCL and FL had split into two clones in the early steps of pathogenesis. This is the first reported case of CL with EBV-negative B-cell non-Hodgkin lymphoma (NHL) and EBV-positive B-cell NHL with a clonal relationship. We discuss the developmental processes of these two lymphomas.

Angiomatous Meningioma in the Craniocervical Junction.

INTRODUCTION: Spinal angiomatous meningioma arising in the craniocervical junction has not been reported. Case Presentation. A 68-year-old man presented to our hospital with pain in the back and left leg. He showed slight motor weakness in his upper extremities. Magnetic resonance imaging revealed a mass with marked enhancement in the craniocervical junction. Computed tomography angiography showed feeding vessels arising from the right vertebral artery. Preoperative embolization of the feeding vessels was performed to reduce intraoperative bleeding. Gross total resection of the tumor was achieved by debulking and piecemeal resection. The tumor attachment to the dura mater was also resected (Simpson grade 1 resection). A histopathological examination confirmed the diagnosis of an angiomatous meningioma. The patient's symptoms improved shortly after surgery. CONCLUSIONS: We achieved gross total resection of spinal angiomatous meningioma arising in the craniocervical junction. A preoperative evaluation and embolization of the feeding arteries may help prevent massive intraoperative bleeding.

Malignant Peripheral Nerve Sheath Tumor of the Cervical Spine Treated with Surgical Resection Followed by X-ray Radiotherapy or Carbon Ion Radiotherapy: A Report of Three Cases.

INTRODUCTION: Spinal malignant peripheral nerve sheath tumors (MPNSTs) are extremely rare. Because of vital surroundings, en bloc resection can be difficult in MPNSTs of the cervical spine. Herein, we report three cases of MPNST followed by radiotherapy or carbon ion radiotherapy (CIRT) after surgery. TECHNICAL NOTE: In case 1, the patient underwent subtotal resection from both a posterior and anterior approach following by adjuvant X-ray radiotherapy. The patient died 13 years after surgery due to liver cancer unrelated to MPNST. In case 2, recurrence spread to the spinal canal in 10 months after primary CIRT. The patient underwent resection of the spinal canal lesion with the residual lesion treated by additional CIRT. Recurrence could be controlled for at least 1 year. In case 3, the patient underwent partial resection for the spinal canal lesion with the residual lesion treated by CIRT. Intradural and extradural recurrences from outside of the CIRT field were observed at 3 years after surgery. CONCLUSIONS: Complete resection and adjuvant X-ray radiotherapy would be an effective treatment for MPNST of the cervical spine, even if en bloc resection with a wide margin is impossible. CIRT for the residual tumor after incomplete resection may have the potential to be an additional treatment option; however, further investigation is warranted.

Paraplegia due to Thoracic Mobile Schwannoma after Myelography.

INTRODUCTION: Spinal mobile tumors are very rare. We herein report a case of paraplegia caused by migration and incarceration of thoracic mobile schwannoma after myelography. Case Presentation. A 25-year-old man who had weakness and numbness in both his legs also had pain radiating to the back that was induced by back flexion or extension and jumping. Magnetic resonance imaging (MRI) showed an intradural extramedullary lesion at the T10 and T11 levels. Myelography was performed but discontinued due to his back and lower limb pain. Computed tomography after myelography revealed a rostrally migrated intradural mass with a discrepancy in the exact location in comparison to the MRI findings. He underwent a second lumbar puncture and drained the cerebrospinal fluid (CSF) to aid the spinal cord, because the symptoms gradually worsened and led to paraplegia. After the drainage of the CSF, his symptoms were immediately resolved. The day after myelography, he underwent complete resection of the tumor with the diagnosis of schwannoma. One year after the surgery, he had been working despite having hyperreflexia in his lower limbs with no weakness or sensory disturbance. CONCLUSION: Severe neurological deficits associated with spinal cord damage can occur due to migration of mobile tumors.

Intradural Extramedullary Spinal Sarcoidosis Mimicking Meningioma.

BACKGROUND: Spinal sarcoidosis is a rare subgroup of neurosarcoidosis. Although most sarcoid lesions develop in the intramedullary compartment, intradural extramedullary (IDEM) spinal sarcoidosis is an extremely rare entity. CASE PRESENTATION: We herein report a case of IDEM spinal sarcoidosis mimicking a meningioma. A 32-year-old man presented to the hospital with clumsy hands and was unable to walk without assistance. Magnetic resonance imaging (MRI) of the cervical spine revealed a dural tail sign that is common in meningiomas. The patient underwent gross total resection. The pathological findings consisted with a sarcoid leision of the spinal cord. The patient's myelopathy recovered after surgery. CONCLUSIONS: Physicians should be alert for the possibility of IDEM sarcoidosis mimicking a meningioma in the differential diagnosis of IDEM spinal cord tumors.

Intradural-Extramedullary Solitary Fibrous Tumor/Hemangiopericytoma with a Negative Result on Fluorodeoxyglucose-Positron Emission Tomography/Computed Tomography.

Intradural-extramedullary solitary fibrous tumor/hemangiopericytoma (SFT/HPC) is a rare entity. SFT/HPCs can recur after surgery, even if a benign histology of the tumor is observed. We herein report a 68-year-old woman with intradural-extramedullary SFT/HPC. On magnetic resonance imaging (MRI), the intradural-extramedullary mass was isointense on T1-weighted images and hypointense on T2-weighted images with heterogeneous gadolinium enhancement. Whole-body fluorodeoxyglucose-positron emission tomography/computed tomography (18F-FDG-PET/CT) was also performed, showing no accumulation. We performed surgery for the intradural-extramedullary mass, and the pathological findings of the resected specimen were a benign histology consistent with World Health Organization (WHO) grade I SFT/HPC. She had no evidence of tumor recurrence three years after the surgery for intradural-extramedullary SFT/HPC. 18F-FDG-PET/CT before surgery may be useful for predicting the postoperative behavior of spinal SFT/HPCs.

Adrenal Ewing's Sarcoma in an Elderly Man.

Ewing's sarcoma usually arises in the bones of children and adolescents. We herein report a 74-year-old man with Ewing's sarcoma in the adrenal gland. The diagnosis was confirmed by a genetic test, pathological studies, and several imaging studies. He already had multiple liver metastases when he was transferred to our hospital and died on the 37th day. The diagnosis was further confirmed by autopsy studies. Adrenal Ewing's sarcoma is very rare, and our patient was older than other reported cases. Ewing's sarcoma should be considered even in elderly patients with adrenal tumors.