Three cases of organized hematoma of the maxillary sinus in patients who underwent preoperative arterial embolization.

Organized hematoma (OH) is benign tumor in the maxillary sinus. The standard treatment for OH is complete surgical resection, however massive bleeding can occur during the procedure, albeit rarely. Some reports have suggested preoperative embolization is useful for reducing the volume of intraoperative bleeding. We report 3 cases of OH in the maxillary performed preoperative embolization. We identified the feeding arteries by angiography or IVR-CT, and we embolized them using Gelatin sponge particles. The embolized artery was the maxillary artery or both the maxillary and the facial artery. There were no major complications as a result of embolization. The mean fluoroscopy time was 35.8 minutes, and the mean fluoroscopy dose was 329.3 mGy. Tumor resection was performed the next day after arterial embolization. The mean bleeding volume for surgery was 383.3 ml, and the mean operative time was 194 minutes. No recurrence was observed in any of the cases over a 4-year follow-up period. We considered that it is possible that preoperative artery embolization is useful for decreasing intraoperative bleeding volume. Although the methods and usefulness of embolization await future reports, it is a technique that should be considered preoperatively because of its potential to prevent massive bleeding.

Various checkpoint molecules, and tumor-infiltrating lymphocytes in common pediatric solid tumors: Possibilities for novel immunotherapy.

Long-term survival rates for pediatric patients with cancer have significantly improved, but novel approaches are desired for those with refractory/relapsed solid tumors. Recently, programed cell death-1/programed cell death-ligand-1 blockade has emerged as an effective option for many intractable cancers. However, not all patients show objective response to such therapy. On the other hand, several other checkpoint pathways, including Herpes virus entry mediator (HVEM)/B- and T-lymphocyte attenuator (BTLA), galectin-9 (GAL9)/T-cell immunoglobulin and mucin domain-3 (TIM3), and major histocompatibility complex class II (MHC-II)/lymphocyte activation gene-3 (LAG3), also regulate immune responses in the tumor microenvironment and may be alternative targets for novel immune therapies. In this study, we examined 65 common pediatric solid tumors and characterized the expression of Herpes virus entry mediator, GAL9, and MHC-II on tumor cells and their corresponding receptors B- and T-lymphocyte attenuator, TIM3, and LAG3, respectively, on tumor-infiltrating lymphocytes (TILs) with immunohistochemistry. Whereas the expression of GAL9 and MHC-II was limited, 73% of rhabdomyosarcomas and 100% of osteosarcomas expressed moderate to high levels of Herpes virus entry mediator on the tumor. TILs were detected in all tumor samples except one osteosarcoma. Interestingly, 45% of rhabdomyosarcomas, and 45% of osteosarcomas expressed moderate to high levels of both Herpes virus entry mediator on the tumor cells and B- and T-lymphocyte attenuator on the TILs. Results showed that a subset of pediatric solid tumors expressed tumor-associated checkpoint molecules, and TILs expressed corresponding receptors for these checkpoint molecules. Thus, immunogenic environments may be created, and checkpoint blockade may induce favorable immune responses.

Behçet disease associated with gastrointestinal amyloidosis manifested as hematochezia: A case report.

RATIONALE: Behçet disease (BD) is an inflammatory disorder characterized by recurrent oral aphthous ulcers, genital ulcers, ocular lesions, and skin lesions. Complication of amyloidosis in patients with BD is rare. Here, we report a case of BD with immunoglobulin light chain (AL)-amyloidosis manifested as hematochezia. PATIENT CONCERNS: A 61-year-old man developed sudden hematochezia due to bleeding from multiple small colonic ulcers; AL-amyloid deposition was found on immunohistochemical examination of biopsy specimen of colonic ulcer. Systemic investigation revealed cardiac disfunction with cardiomegaly and progressive renal dysfunction, which indicated the presence of systemic AL-amyloidosis. DIAGNOSES: Based on the findings of colonic ulcers with cardiac and renal involvement, a diagnosis of systemic AL-amyloidosis complicated by incomplete BD was established. INTERVENTIONS: He was treated with increased dose of oral prednisolone (20 mg/day), colchicine and mesalazine, because he was reluctant to receive aggressive chemotherapy (melphalan and dexamethasone) or autologous stem cell transplantation. OUTCOMES: Colonic ulcers completely diminished after treatment, however, he died because of severe urinary tract infection and progressive renal failure after one year of gastrointestinal (GI) manifestations. LESSONS: Our case shows that patients with BD may have GI manifestations due not only to entero-BD but also due to GI amyloidosis.

A Case of Squamous Cell Carcinoma of Unknown Primary that Responded to the Multi-Tyrosine Kinase Inhibitor Lenvatinib.

Lenvatinib is an oral tyrosine kinase inhibitor of vascular endothelial growth factor receptors 1, 2, and 3, fibroblast growth factor receptors 1 through 4, as well as platelet-derived growth factor receptor α, RET, and KIT. At present, lenvatinib is used in the treatment of thyroid cancer and renal cell carcinoma. We herein report a case of a 67-year-old patient with squamous cell carcinoma of unknown primary who was effectively treated with lenvatinib. The patient was initially diagnosed as having undifferentiated thyroid cancer, and after total thyroidectomy and bilateral lymph node dissection, lenvatinib was administered for the treatment of residual lymph node metastasis. A computed tomography scan after 1 month of lenvatinib administration showed marked regression of the lymph nodes, but interstitial pneumonia was also detected. Because the drug lymphocyte stimulation test for lenvatinib was strongly positive, we concluded that the interstitial pneumonia was induced by lenvatinib. The interstitial pneumonia only improved by the withdrawal of lenvatinib. Finally, his thyroid tumor was diagnosed as a metastasis of squamous cell carcinoma; however, we were unable to identify the primary lesion. This is the first reported case of interstitial pneumonia induced by lenvatinib.

Contrast uptake in primary hepatic angiosarcoma on gadolinium-ethoxybenzyl-diethylenetriamine pentaacetic acid-enhanced magnetic resonance imaging in the hepatobiliary phase.

Primary hepatic angiosarcoma is the most common malignant mesenchymal tumor of the liver. It has a poor prognosis and various appearances on magnetic resonance (MR) images. We report a case of hepatic angiosarcoma with a characteristic appearance on gadolinium-ethoxybenzyl-diethylenetriamine pentaacetic acid (Gd-EOB-DTPA)-enhanced MR imaging in the hepatobiliary phase. A 72-year-old man was admitted with a complaint of abdominal pain. Gd-EOB-DTPA-enhanced MR imaging revealed a liver tumor that showed slight hyperintensity in the hepatobiliary phase. These findings suggested Gd-EOB-DTPA uptake in the tumor. An autopsy revealed the solid proliferation and sinusoidal spreading of hepatic angiosarcoma cells. Immunohistochemistry indicated that the tumor was negative for OATP1B3. Gd-EOB-DTPA uptake in the liver tumor in the hepatobiliary phase suggested sinusoidal tumor invasion with residual normal hepatocytes.

A case of gastric lipoma resected by endoscopic submucosa dissection with difficulty in preoperative diagnosis.

A 66-year-old man was referred to our hospital with an increasing subepithelial lesion in the gastric antrum. Using esophagogastroduodenoscopy, a tumor with a steep, 20-mm-high rise protruding in the lumen was observed. The mucosal surface of the tumor was reddish, with ulcers forming at the base. Moreover, the tumor was mobile and soft. A biopsy specimen was taken from the ulcer, but tumor tissue was not collected from the submucosa. Endoscopic ultrasonography (EUS) showed a high echoic mass in the submucosa. However, because the mucosal surface of the ulceration was red, the mesenchymal tumor with internal bleeding was inferred to be lipoma. Additionally, because the tumor was small, flexible, and soft, collecting tumor tissue under EUS-guided fine-needle aspiration was inferred as difficult. We were unable to make a final diagnosis because the lesion showed a small tumor with atypical macroscopic morphology. Therefore, endoscopic submucosa dissection (ESD) was chosen for the diagnostic treatment. Sodium hyaluronate sufficient for separation from the muscular layer was injected into the submucosa. Then submucosal dissection was performed just above the muscle layer. Results demonstrate the possibility of removing the tumor reliably without perforation. Pathological evaluation of the ESD specimen indicated a diagnosis of gastric lipoma.

Primary malignant lymphoma of the talus: A case report and review of the literature.

Malignant lymphoma commonly occurs in adults, with a peak incidence between the seventh and ninth decades of life. Although malignant lymphoma usually occurs in the lymph nodes, it rarely occurs primarily in the bone. We herein describe an extremely rare case of primary malignant lymphoma of the talus in a 74-year-old man. Although plain radiographs showed no abnormality, gadolinium-enhanced magnetic resonance imaging (MRI) revealed a well-circumscribed intra- and extraosseous tumor of the talus. 18F-fluorodeoxyglucose (FDG) positron emission tomography/MRI revealed a marked increase of FDG uptake in the right ankle and the right inguinal lymph nodes. As the tumor was diagnosed as diffuse large B-cell lymphoma by core needle biopsy, combination therapy with chemo- and radiotherapy was initiated. The patient achieved complete remission, with no sign of recurrence at 8 months after initial chemoradiotherapy. Since primary malignant lymphoma of the bone is chemo- and radiosensitive and has a good prognosis, accurate staging by radiological investigation as well as correct pathological diagnosis by biopsy are required for optimal treatment.

Pancreatic neuroendocrine tumor Grade 1 patients followed up without surgery: Case series.

Among the three grades of neuroendocrine tumors (NETs), the prognosis for Grade 1 (G1) with surgery is very good. Therefore, we evaluated the prognoses of pancreatic NET (PNET) G1 patients without surgery. A total of 8 patients who were diagnosed with NET G1, with an observation period of more than 6 mo until surgery or without surgery, were recruited. The patients who underwent surgery were ultimately diagnosed using specimens obtained during the surgery, whereas the patients who did not undergo surgery were diagnosed using specimens obtained by endoscopic ultrasonography-guided fine needle aspiration. Overall, we mainly evaluated the observation period and tumor growth. The observation period for the five cases with surgery ranged from 6-80 mo, and tumor growth was observed in one case. In contrast, the observation period for the three cases without surgery ranged from 17-54 mo, and tumor growth was not observed. Therefore, although the first-choice treatment for NETs is surgery, our experience includes certain NET G1 patients who were followed up without surgery.

[A Case of Primary Poorly Differentiated/Small Cell Carcinoma of the Breast in a Patient with Von Recklinghausen's Disease].

We report a case of neuroendocrine carcinoma and poorly differentiated/small cell carcinoma ofthe breast in a patient with von Recklinghausen's disease. The patient was a 46-year-old woman who was diagnosed with von Recklinghausen's disease when she was 22 years old. She presented with left breast pain, and physical examination revealed a firm mass in the left breast. A core needle biopsy of the tumor revealed triple negative breast cancer with neuroendocrine features. We performed a simple mastectomy with lymph node dissection. We did not plan neoadjuvant chemotherapy because the tumor would be possibly inoperative if neoadjuvant chemotherapy was not effective for this neuroendocrine cancer. The tumor was diagnosed as a neuroendocrine carcinoma and poorly differentiated/small cell carcinoma. The patient was treated with CDDP and CPT- 11, which is a regimen often used to treat small cell lung cancer.

[Surgical Resection for a Metachronous Liver Metastasis of an Esophagogastric Junction Adenocarcinoma].

The patient was a 56-year-old man with advanced esophagogastric junction cancer. He received neoadjuvant chemotherapy with 5-FU plus CDDP followed by lower esophagectomy and total gastrectomy via the left thoracoabdominal approach in October 2011. Pathological examination revealed EGJ adenocarcinoma (ypT4aN1M0, Stage ⅢA, Japanese Classification of Gastric Carcinoma ver.14), and histological analysis indicated Grade 0 (no change). Adjuvant chemotherapy with S-1 was administered. Nevertheless, 6 months after the operation, a solitary hepatic metastasis (f: 32 mm) was detected in S7 of the liver. The patient underwent proton beam irradiation of the liver metastasis, resulting in a complete response, and he was followed up without any chemotherapy. However, 21 months after the irradiation, regrowth of the previous lesion with FDG re-accumulation was noted. Given the absence of any neoplasms other than the liver metastasis, right hepatic lobectomy was performed. Pathological examination revealed a small cluster of viable tumor cells surrounded by extensive fibrotic tissue (Grade 2). At 45 months after the initial operation (10 months after the liver lobectomy), the patient is living without any signs of recurrence. Surgical resection for liver metastasis of EGJ cancer may be feasible after careful selection.