A case of adenomyosis with leiomyoma that was effectively treated with relugolix and kamishoyosan add-on therapy.

BACKGROUND: Recently, relugolix, an oral gonadotropin-releasing hormone receptor antagonist, has been considered an effective therapy for leiomyoma based on a phase 3 study in Japanese women. Leiomyoma combined with severe adenomyosis occasionally occurs in perimenopausal women; however, little information on the effectiveness of relugolix against severe adenomyosis exists. CASE PRESENTATION: A 49-year-old woman was referred to our hospital with acute lower abdominal pain and abnormal uterine bleeding. Magnetic resonance imaging revealed multiple leiomyomas with diffuse adenomyosis. Left hydrosalpinx was also observed. The patient refused surgical treatment and preferred oral relugolix. Since she experienced a hot flush and headache induced by relugolix, a traditional Japanese Kampo, kamishoyosan, was added to improve the side effects of relugolix. The patient was asymptomatic at the time of this report and experienced a significant shrinkage in uterine volume. Ultimately, she avoided hysterectomy as desired. CONCLUSIONS: To our knowledge, this is the first report of co-occurring adenomyosis and leiomyoma, which was effectively treated with relugolix. Although the management of adverse side effects, including hot flush and headache by relugolix, has recently attracted attention and controversy, relugolix add-on therapy with kamishoyosan may help treat menopausal symptoms.

A fat containing combined neuroendocrine carcinoma and hepatocellular carcinoma in the liver: A case report.

A 79-year-old man was admitted to our hospital because of increased hepatobiliary enzyme levels. Dynamic computed tomography and magnetic resonance imaging showed a liver tumor measuring 60mm containing fat foci at the cranial aspect of the tumor. We diagnosed the patient with hypovascular hepatocellular carcinoma (HCC) and fat deposition, and performed a caudate lobe resection. Pathology examination revealed two intermingled components: moderately differentiated HCC with fat deposition and neuroendocrine carcinoma (NEC). Primary combined NEC and HCC is extremely rare. To our knowledge, this is the first report of combined NEC and HCC including a fat component. HCC is the most common primary hepatic malignancy with fat. HCC might include fat, even if HCC coexists with another type of cancer. The imaging characteristics of and HCC with another type of cancer vary depending on the amount of each component. We should not simply diagnose such tumors as HCC, but think about the possibilities of HCC with another type of cancer, because there is a fat component.

Clinical, Morphologic, and Pathologic Features Associated With Increased FDG Uptake in Schwannoma.

OBJECTIVE: The objective of this study was to investigate the clinical, morphologic, and pathologic features associated with increased 18F-FDG uptake in benign schwannomas. MATERIALS AND METHODS: Twenty-two schwannomas in 22 patients (age range, 25-81 years) who had FDG PET or PET/CT scans and subsequently underwent surgical re-section were retrospectively analyzed. The maximum standardized uptake value (SUVmax) was compared with patient age, sex, tumor location (gastrointestinal vs nongastrointestinal origin), tumor size, gross appearance, intratumoral cellularity, intratumoral infiltration of inflammatory cells, presence of peritumoral lymphoid cuffs, and expression status of glucose transporters 1 and 3 on tumor cells. RESULTS: The SUVmax of schwannomas ranged from 1.5 to 17.3 (median, 3.7). Significantly higher SUVmax was observed in gastrointestinal schwannomas (n = 4) compared with nongastrointestinal schwannomas (n = 18, p = 0.007) and in schwannomas with peritumoral lymphoid cuffs (n = 5) compared with those without peritumoral lymphoid cuffs (n = 17, p = 0.001). A significant correlation was seen between tumor location and the presence of peritumoral lymphoid cuffs (p < 0.001). Age, sex, tumor size, gross appearance, intratumoral cellularity, intratumoral inflammatory cell infiltration, and expression status of glucose transporters 1 and 3 on tumor cells had no significant correlation with SUVmax. CONCLUSION: Gastrointestinal schwannomas and schwannomas with peritumoral lymphoid cuffs may be associated with elevated FDG uptake. Knowledge of the features of schwannomas associated with increased uptake may be helpful to avoid misinterpretation of benign schwannomas as malignancy.

Invasive cervical cancer accompanied by IgG4-related disease.

IgG4-related disease (IgG4-RD) is a systemic disease that affects multiple organs and generates nodules or thickening. Discriminating these diseases from malignancy is important because glucocorticoid treatment is effective for patients with IgG4-RD. Coexistence of IgG4-RD with various malignant diseases has been reported, but there are few reports with regard to gynecologic malignant diseases. We encountered a case of invasive cervical cancer stage IIB accompanied by IgG4-RD. The patient was a 46-year-old woman. On pelvic magnetic resonance imaging, fluorodeoxyglucose-positron emission tomography and computed tomography, systemic multiple lymph node swelling was seen, including in the neck and the mediastinum in addition to uterine cervix. Diagnosis (and hence, appropriate treatment choice) was achieved on pathology of the submandibular gland and uterus, and analysis of serum IgG4. IgG4-RD should be suspected in patients presenting with malignancy and unusual multiple lymph node swelling.

[The Combination Therapy of Gemcitabine Plus Paclitaxel Induced Complete Response in a Tongue Skin Brain Metastasis of Bladder Carcinoma: A Case Report].

Tongue, skin and brain metastases of bladder cancer are very rare and few cases have been reported. We report a case of tongue, skin and brain metastases of bladder cancer. A 61-year-old woman was referred to our hospital with gross hematuria. Transurethral resection of the bladder (TURBT), tongue biopsy and skin biopsy were performed. Pathological findings showed urothelial carcinoma, G2, micro papillary variant, pT2＞ and tongue and skin metastases from urothelial carcinoma of bladder. After three cycles of chemotherapy (gemcitabine plus paclitaxel), tongue and skin metastases disappeared. Cystoscopy revealed no tumor of bladder. Eleven months later, she was admitted to our hospital because of disturbance of consciousness. Magnetic resonance imaging (MRI) showed multiple brain metastases. Rechallenge of chemotherapy (gemcitabine plus paclitaxel) restored from disturbance of consciousness and MRI showed partial response of brain metastases. We performed six additional courses of chemotherapy. Skin, tongue and brain metastases from bladder cancer indicate poor risk. Chemotherapy (gemcitabine plus paclitaxel)could be effective against these matastases.

[Case of advanced colon cancer after colostomy closure].

We report a case of advanced colon cancer after closure of a colostomy. A 77-year-old man suddenly began experiencing abdominal pain and pan-peritonitis due to perforate diverticulum of the sigmoid colon. A Hartmann operation was urgently performed, and closure of the colostomy was performed 6 months later. The patient had no history of colonoscopy. Colonoscopy was performed 45 months after the closure of the colostomy to examine the colon. Advanced colon cancer was detected near the anastomotic region. In conclusion, older patients, including those with numerous disease complications such as diabetes and hypertension, should undergo early colonoscopy as a preventative measure.

[A case of acute subdural hematoma from dissection of cortico-dural anastomosis].

A case of acute subdural hematoma caused by rupture of a bridging artery between the dura and the cortical surface is reported. A 49-year-old man complained of headache in the left temporal area after he had lifted up a heavy load. His symptom did not improve in spite of taking analgesics, so he was transferred to our hospital. Brain CT disclosed left acute subdural hematoma. Soon after admission, he became comatose and developed right hemiplegia. The cerebral angiogram displayed a tapering occlusion of a branch of the left middle cerebral artery. Emergent decompressive craniectomy was performed, and the anastomotic artery between the dura mater and the cortical artery was followed until it appeared normal in the cortical sulcus. Proximal occlusion of the artery was carried out. Pathological examination demonstrated the dissection of the anastomotic artery. This is the first reported and pathologically-proven case of acute subdural hematoma caused by dissection of the anastomotic artery between the dura mater and the cortical artery.

Fatal hemoperitoneum due to rupture of the left gastric artery in a patient with microscopic polyangiitis.

Microscopic polyangiitis is a vasculitis which primarily affects capillaries, venules or arterioles. Involvement of small and medium-sized arteries may also occur. A 70-year-old Japanese female with a fever and cough was diagnosed with pneumonia and antibiotics were administered. Her symptoms initially improved, but her fever recurred and she experienced malaise and loss of appetite. Her renal function gradually worsened and she was positive for myeloperoxidase antineutrophil cytoplasmic antibodies (MPO-ANCA). She was referred to our hospital on the suspicion of ANCA-associated glomerulonephritis. However, her depressive mental symptoms did not allow her to undergo a renal biopsy. She was clinically diagnosed with ANCA-associated glomerulonephritis, and oral corticosteroids and intravenous methylprednisolone were administered. Her symptoms and renal function were improved, but she died suddenly 15 days after admission. An autopsy disclosed approximately 700 mL bloody ascites. Coagulation adhered to the lesser curvature of the stomach, but the source of hemorrhage could not be detected macroscopically because the gastric mucosa did not show abnormal findings. The histological findings revealed that the left gastric artery showed necrotizing angiitis and rupture. In the kidneys, cellular crescents were found in approximately 10%, fibrous crescents were found in approximately 10%, sclerosis and collapse were found approximately 30% of the glomeruli, and necrotizing angiitis was observed in interlobular arteries and arterioles. From these findings, she was finally diagnosed with microscopic polyangiitis. Microscopic polyangiitis is an extremely rare cause of spontaneous intraperitoneal bleeding, but it must be carefully considered in the differential diagnosis for the appropriate management of such patients.

[Acquired hemophilia A developing at bilateral renal bleeding: a case report].

A 82-year-old woman with asymptomatic gross hematuria was referred to us for a consultation. Computed tomography and magnetic resonance imaging revealed hydronephrosis in the right kidney and retrograde pyelography showed a filling defect in the right renal pelvis. Cystoscopy revealed gross hematuria from right ureteral orifice concomitant with superficial papillary bladder tumor (catheterized urine cytology: positive) (transurethral resection of bladder tumor UC G1 pTa). She was diagnosed with right renal pelvic carcinoma, and we performed a total right nephroureterectomy. Histolopathological findings showed no evidence of malignancy, but extensive hemorrhaging was observed in the subepithelium. There was hematoma in the retoroperitoneum and she suffered from endotoxic shock after the operation. In the post-operative period, she showed complications involving left renal bleeding. She was diagnosed with acquired hemophilia A based on a decline in factor VIII activity, prolonged APTT and the presence of anticoagulant. Although we administered steroids to her, her general condition deteriated and she died of postrenal renal failure 37 days after the operation. To our knowledge, this is the fourth case worldwide and the third case in Japan of acquired hemophilia A discovered by renal bleeding.

Repeated Short-Term Recurrence of Chronic Subdural Hematoma Associated with Metastasis to Hematoma Capsule Originating from Extracranial Malignant Tumor.

BACKGROUND: The recurrence rate of chronic subdural hematoma (CSDH) after trepanation is relatively high and involves various factors. We encountered an extremely rare case in which metastasis of an extracranial malignant tumor to the hematoma capsule was thought to be causing short-term repeated recurrences of CSDH. CASE DESCRIPTION: The patient was a 74-year-old man who had undergone bur hole evacuation of left CSDH 7 months earlier and had been receiving chemotherapy for stage IV gastric cancer for the preceding 6 months. He presented with symptoms of right hemiparesis and was diagnosed with left CSDH. Bur hole evacuation was performed twice, but the hematoma enlarged again both times within a short period. A third bur hole evacuation was performed after middle meningeal artery embolization, but the hematoma again enlarged shortly thereafter. Hematoma enlargement was finally suppressed by extracting the hematoma capsule under craniotomy. In histopathologic examinations, hematoxylin-eosin staining showed poorly differentiated adenocarcinoma primarily along the luminal side of the hematoma capsule and immunohistochemical staining showed results identical to findings from the existing gastric cancer. Pathologic results confirmed metastasis of the gastric cancer to the hematoma capsule, and this was considered to be the cause of short-term repeated recurrence of CSDH. CONCLUSIONS: To the best of our knowledge, this is the first report of metastasis to a hematoma capsule originating from an extracranial malignant tumor. The clinical course in the present case suggests metastasis of extracranial malignant tumor to the CSDH capsule as an extremely rare cause of recurrence.

Small cell lung cancer transformation during immunotherapy with nivolumab: A case report.

We report a rare case of transformation of non-small cell lung cancer (NSCLC) to small cell lung cancer (SCLC), without epidermal growth factor receptor (EGFR) gene mutation, during immunotherapy treatment with nivolumab. A 75-year-old man was referred to our hospital following the observation of a 64 mm mass in a chest computed tomography (CT) scan. A transbronchial biopsy of the mass identified the pathological presence of poorly differentiated NSCLC, with no histological signs of SCLC. No mutations were identified in the EGFR gene. A clinical diagnosis of NSCLC (cT3N3M1a, stage IV) was made following a positron emission tomography (PET)-CT scan and enhanced brain magnetic resonance imaging. Docetaxel and bevacizumab were selected as the first-line chemotherapy regimen; however, after two cycles, the patient developed a gastrointestinal perforation, and discontinuation of cytotoxic chemotherapy was recommended. Owing to gradual disease progression, immunotherapy with nivolumab was selected as the second-line regimen. During the immunotherapy, the tumor continued to progress and some subcutaneous tumors emerged. Biopsy of a subcutaneous tumor revealed SCLC, with positive immunostaining for cluster of differentiation 56, synaptophysin, and thyroid transcription factor-1. Serum tumor markers of SCLC were also elevated. Based on these results, we concluded that in this case NSCLC had transformed to SCLC during immunotherapy with nivolumab.

Anti-PD1 Antibody Treatment and the Development of Acute Pulmonary Tuberculosis.

Recently, cancer immunotherapy by immune checkpoint inhibitors has been considered one of the pillars for the treatment of cancer. Nivolumab is the first immune checkpoint inhibitor approved for lung cancer treatment in Japan. Although nivolumab has superior survival benefits and fewer adverse events than cytotoxic agents, it can generate dysimmune toxicities, known as immune-related adverse events. Although autoimmune manifestations are well-known immune-related adverse events, the development of infectious diseases is rare. Here, we report on a patient with advanced NSCLC in whom pulmonary tuberculosis developed rapidly during nivolumab treatment and discuss the potential mechanisms as well as what is known about infections during checkpoint inhibitor therapy.

Giant mature teratoma in the mediastinum presenting with rapid growth.

Teratomas are primary germ-cell tumours in the mediastinum. Although they are generally slow-growing and asymptomatic, rapid growth causing life-threatening complications can occur. Sebaceous secretion, insulin production, chorionic gonadotropin secretion and pancreatic enzyme secretion are the presumptive causes of tumour progression. Only few cases of rapidly growing teratomas have been reported previously. Here, we present a case of a giant mature teratoma in the mediastinum that presented with rapid growth and compare the characteristics of this case with those of previous cases.