[False-Positive ¹²³I-Metaiodobenzylguanidine Scan in a Patient with Adrenocortical Cancer].

A 59-year-old woman presented with a left adrenal tumor 4 cm in diameter. The ¹²³I-metaiodobenzylguanidine (MIBG) scintigraphy showed apparent accumulation in the left adrenal tumor. However, the patient had no sign or symptoms suggesting pheochromocytoma. No biochemical evidence of catecholamine excess was noticed. Computed tomography (CT) revealed relatively strong enhancement in the arterial phase, which persisted until the portal phase. The computed tomography (CT) and magnetic resonance imaging showed 2 liver nodule suspected to be metastatic tumors. No ¹²³I-MIBG accumulation was seen in these nodules. CT also showed thyroid nodules with calcification, which suggested papillary thyroid cancer. Based on the findings, open left adrenalectomy, partial hepatectomy and segmentectomy were performed under the clinical diagnosis of pheochromocytoma and metastatic liver tumors. Histopathological diagnosis was adrenocortical cancer. There was only lymphocyte infiltration in the liver nodules. Postoperative recovery was uneventful, and the patient underwent partial thyroidectomy 6 months later. The pathological diagnosis was papillary thyroid cancer. She has been without recurrence or metastases for 18 months after adrenalectomy. We found only 6 cases of MIBG scintigraphy-positive adrenocortical cancer in the literature. The mechanisms for MIBG uptake in adrenocortical cancer are discussed with a review of the literature.

[A Case of Retroperitoneal Bronchogenic Cyst Successfully Resected with Retroperitoneoscopic Surgery].

A 32-year-old woman was referred to our hospital for consultation with a suspected left adrenal tumor detected by ultrasonography at a health check. Computed tomography and magnetic resonance imaging revealed a 3×1×4 cm multilocular cystic mass arising from the diaphragmatic crura, suggesting a retroperitoneal bronchogenic cyst. The patient underwent excision of the cyst and adjacent diaphragm using a retroperitoneoscopic approach. Retroperitoneal bronchogenic cysts are rare. We review the cases of 26 patients who underwent laparoscopic resection of a retroperitoneal bronchogenic cyst.

High prevalence of hypogonadism determined by serum free testosterone level in Japanese testicular cancer survivors.

OBJECTIVE: Hypogonadism is a major complication in testicular cancer survivors, but its prevalence varies among studies. In Japan, free testosterone has been used for diagnosis of late-onset hypogonadism syndrome. In the present study, we evaluated the hormone level of testicular cancer survivors and its impact on their quality of life. METHODS: Overall, 50 testicular cancer survivors treated from 1990 to 2013 were enrolled. The median age was 44 years. The serum levels of free testosterone, total testosterone and luteinizing hormone were measured. All patients completed the Aging Males' Symptom scale and International Index of Erectile Function-15. The hormone levels of 337 healthy volunteers were used as the control. RESULTS: A total of 32 (64%) patients showed free testosterone levels <8.5 pg/mL. In contrast, just 26% of 50 patients showed total testosterone levels <3.5 ng/mL. Testicular cancer survivors had significantly lower free testosterone and higher luteinizing hormone compared with healthy controls. In contrast, there was no difference in total testosterone between patients and controls. The prevalence of late-onset hypogonadism symptoms of any grade (Aging Males' Symptom total score ≥27) was 60%. Overall, 64% were defined as having moderate erectile dysfunction (International Index of Erectile Function-Erectile Function domain score <17). However, Aging Males' Symptom, International Index of Erectile Function-15 and Erectile Function domain scores did not differ by free testosterone or total testosterone level. CONCLUSIONS: This is the first report on the prevalence of hypogonadism determined by free testosterone level in Japanese testicular cancer survivors. Because Aging Males' Symptom and International Index of Erectile Function-15 scores do not necessarily reflect the hormone level, measuring free testosterone is also important in the follow up of these patients.

[A Case of Upper Urinary Tract MALT Lymphoma with Remarkable Thickness of Renal Pelvis and Ureter Wall].

A 78-year-old man was referred to Tsukuba University Hospital for right hydronephrosis. He had undergone ureteroscopy and ureteral stenting in another hospital, but no tumor was revealed in renal pelvis and ureter. The urinary cytology was negative. Computed tomography (CT) revealed remarkable thickening of right renal pelvis and ureter wall. CT also showed para-aortic, iliac, supraclavicular and mediastinal lymph node (LN) swelling. 18F-fluoro-2-deoxy-D-glucose positron emission tomography (PET) revealed high uptake at thickened right renal pelvis and ureter wall and enlarged LNs. The soluble interleukin-2 receptor was elevated to 1,110 U/ml (normal range: 613 U/ml). Those findings suggested that the malignant lymphoma originated from the renal pelvis and ureter rather than urothelial cancer. Therefore we performed open biopsy of iliac LN and periureteral tissue. The pathological diagnosis was mucosa associated lymphoid tissue (MALT) lymphoma. The patient was trasferred to the department of hematology, and treated with rituximab and bendamustine. After 6 courses of chemotherapy, swelling of renal pelvis, ureter and LN was markedly reduced. The ureteral sent could be removed. MALT lymphoma of the upper urinary tract is extremely rare and pretreatment diagnosis is difficult. In 8 of 11 reported cases, the diagnosis was made by nephroureterectomy. In our cases, open biopsy could avoid nephroureterectomy.

[Metachronous Development of Mantle Cell Lymphoma during Active Surveillance in a Patient with Stage I Testicular Cancer].

A 78-year-old man presented with swelling of para-aortic and left iliac lymph nodes (LNs). He had undergone left high orchiectomy 10 years ago. He was diagnosed with stage I seminoma, and was managed with active surveillance. Eight years later, the follow-up computed tomography (CT) revealed para-aortic LN swelling, but the patient refused further treatment. He complained of left lower extremity edema 10 years after orchiectomy. CT showed enlargement of both LNs, especially, diameter of left iliac lymph nodes was up to 9 cm. He was referred to our hospital. LDH was slightly increased to 261 IU/1, but α-fetoprotein, and total and free beta human chorionic gonadotropin were within normal limits. Results of pathological review of the testicular tumor was also seminoma. The treatment with etoposide and cisplatin (EP) was started under the diagnosis of late relapse of seminoma. However, CT after 1 course of EP showed no shrinkage of LN metastases. At this time, soluble interleukin-2 receptor (sIL-2R) was elevated up to 5,090 U/ml (normal range: 613 U/ml). Needle biopsy from pelvic LN was performed on suspicion of metachronous malignant lymphoma. The pathological diagnosis was mantle cell lymphoma. The patient was transferred to the department of hematology, and treated successfully with rituximab and bendamustine. When LN swelling is seen after long-term active surveillance, there is a possibility of late relapse. However, metachronous malignant lymphoma also should be considered in an elderly patient.

[A Case of Leiomyosarcoma Arising from the Ovarian Vein Preoperatively Diagnosed with Radiological Imaging].

A 54-year-old woman presented withtransient back pain. She was diagnosed withleiomyosarcoma of the inferior vena cava (IVC) by computed tomography (CT) and was referred to our hospital. Contrastenhanced CT revealed a mass (38×42 mm) located in the retroperitoneal space along the course of the right ovarian vein. The mass compressed the IVC into a crescent shape. A tumor thrombus was also found in the IVC. 18 F-fluoro-2-deoxy-D-glucose positron emission tomography (PET) revealed high uptake at the caudal side of the tumor. These radiological findings strongly suggested the diagnosis of leiomyosarcoma arising from the right ovarian vein. She underwent tumor resection with right nephrectomy, IVC resection, and IVC patch reconstruction without any notable events after surgery. Histopathological diagnosis was leiomyosarcoma arising from the ovarian vein, not from the IVC. Two months after the surgery, CT revealed multiple pulmonary metastases and a single liver metastasis. The patient was referred to another hospital for further treatment. She was treated with chemotherapy and was alive with disease at 14 months after the surgery.

[A Case of Primary Schwannoma of the Urinary Bladder].

A 68-year-old woman presented with a bladder tumor. She was asymptomatic, and the tumor was incidentally detected with radiological imaging performed during treatment of cervical cancer. Magnetic resonance imaging and computed tomography revealed a solitary submucosal tumor located in the anterior wall of the urinary bladder, with homogeneous contrast enhancement. Cystoscopy showed a submucosal tumor covered by normal mucosa. A paraganglioma was considered in the differential diagnosis, but symptoms suggesting hypercatecholaminemia were not apparent. Moreover, she did not have a family history or symptoms associated with neurofibromatosis-1 (NF-1). She underwent partial cystectomy with a preliminary diagnosis of submucosal bladder tumor. Histopathological diagnosis confirmed a schwannoma arising from the bladder wall. She was followed up without intravesical recurrence or metastases for 6 months. In the literature, only 12 cases of bladder schwannoma have been reported. There was no reported family history or symptoms associated with NF-1 in any of the cases. Although the number of cases is limited, literature review showed a favorable prognosis for bladder schwannoma with local tumor resection in patients without NF-1.

[Screening of Deep Vein Thrombosis Prior to Surgery Using D-dimer Testing and Venous Ultrasonography of Lower Extremities].

Deep vein thrombus (DVT) in a patient awaiting surgery is a considerable source of pulmonary embolisms (PEs) during the surgical period, but screening for DVTs has not been emphasized. This retrospective study was conducted to identify factors associated with a positive D-dimer result and DVT diagnosis in order to evaluate the usefulness of DVT screening for patients awaiting surgery. A total of 1,061 patients (872 males, 189 females) underwent D-dimer testing prior to urological surgery at Tsukuba University Hospital between April 2013 and March 2016. Factors associated with a positive D-dimer result and DVT diagnosis were determined by a univariate analysis. Among the 75 patients with a positive Ddimer result, venousultras onography of the lower extremitieswasperformed in 69 patientsand DVT was diagnosed in 14 patients. The overall true-positive rate of D-dimer was 20. 3% (14/69), and it was significantly higher in the females (males11.3% vsfemales50% ; p＝0.0021). Age, body mass index and Ddimer value were not associated with the true-positive D-dimer result. Among the 1,061 patients, DVT was significantly more likely to be diagnosed in elderly patients (median age 73.5 vs 67 yrs, p＝0.0087) and females(males0. 69% vsfemales4. 23% ; p＝0. 0010). Among the three patientswith an acute-phase thrombus, two postponed surgery and required anti-thrombus therapy, and the other patient underwent the implantation of an inferior vena cava filter in order to undergo surgery on schedule. No PE occurred during the surgical period. These results indicate that DVT screening for patients awaiting surgery is useful and should be considered as part of the prevention of PEs during the surgical period.

[Two Cases of Germ Cell Tumors with Hyperthyroidism Due to High Serum hCGLevels].

We reported two cases of hyperthyroidism that developed during induction chemotherapy for advanced germ cell tumors with high serum human chorionic gonadotropin (hCG) levels. Case 1 : An 18-year-old man with mediastinal choriocarcinoma complained of tachycardia and tremor. His pretreatment serum hCG level was 1.37 million mIU/ml. The free thyroxine (fT4) level measured on day 2 of the first course of bleomycin, etoposide and cisplatin (BEP) was elevated to 7.8 ng/dl (＜1.7 ng/dl), whereasthe thyroidstimulating hormone (TSH) level was undetectable. We diagnosed the patient with hyperthyroidism and started oral propranolol and thiamazole. Subsequently, his tachycardia and tremor disappeared. On day 12 of the first course of BEP, his hCG level decreased to less than 50,000 mIU/ml. Also, his fT4 level returned to the normal range. Case 2 : A 29-year-old man presented with a left scrotal mass. He was diagnosed with non-seminoma testicular cancer (embryonal carcinoma and choriocarcinoma) with multiple lung, liver and lymph node metastases. On the admission day, his serum hCG and fT4 levels were high ; 3.23 million mIU/ml and 2.2 ng/dl, respectively. The TSH level was low at 0.011 mIU/ml. On day 3 of the first course of BEP, his hCG and fT4 levels increased to 4.5 million mIU/ml and 3.0 ng/dl, respectively. He complained of tachycardia, tremor and hyperhydrosis. He was started on propranolol and potassium iodide. After the treatment, histachycardia, tremor and hyperhidrosisdis appeared. HisfT4 level normalized on day 17 of the first course of BEP. The TSH-like activity of hCG is considered to be responsible for paraneoplastic hyperthyroidism among germ cell cancer patients with high hCG levels. To our knowledge, thisisthe first report of such a case in Japan. However, thisphenomenon isnot rare among patients with extremely high hCG levels. Therefore, we should be careful of these patients.

[Incidence and Risk Assessment of Tumor Lysis Syndrome in Patients with Advanced Germ Cell Cancer].

Tumor lysis syndrome (TLS) is a major oncological emergency. TLS is common in patients with hematological malignancies, but it can occur across a spectrum of cancer types. Germ cell tumors (GCT) have rapid cancer cell turnover and often present with bulky metastasis. The international TLS expert consensus panel has recommended guidelines for a medical decision tree to assign low, intermediate and high risk to patients with cancer at risk for TLS. GCT is classified as intermediate risk for TLS, and the patients who have other TLS risks factors are classified to be at high risk for TLS. In this study, we retrospectively analyzed 67 patients with metastatic GCT who were treated with induction chemotherapy at Tsukuba University Hospital between 2000 and 2013. Thirty-one, 15 and 21 patients were classified with good-, intermediate- and poor-prognosis disease, respectively, according to the International Germ Cell Cancer Collaborative Group criteria. Twelve patients (18%) were classified to be at high risk for TLS, and two patients were treated with allopurinol or rasburicase as prophylaxes for TLS. They did not show progression to laboratory TLS (L-TLS). In the remaining 10 TLS high-risk patients, three (30%) patients developed L-TLS after chemotherapy and started receiving oral allopurinol. As a result, no patients developed clinical TLS (C-TLS). In this study, 30% of TLS-high risk patients developed L-TLS without prophylactic treatment. Therefore, it is important to conduct TLS-risk stratification and consider prophylaxis such as rasburicase for advanced GCT patients at induction chemotherapy.

A case of primary ciliary dyskinesia treated with ICSI using testicular spermatozoa: case report and a review of the literature.

Purpose: To investigate whether or not intracytoplasmic sperm injection (ICSI) using spermatozoa extracted from testis (TESE-ICSI) is a more effective treatment than ICSI with ejaculated spermatozoa (EJ-ICSI) for primary ciliary dyskinesia (PCD). Methods: We reported a case of PCD in which we performed TESE-ICSI after repeated failure of EJ-ICSI. Together with data from previous case reports, we compared the fertilization rate and pregnancy outcome of TESE-ICSI and EJ-ICSI. Results: In our case, TESE-ICSI improved the morphology of spermatozoa and fertilization rate. However, the outcome was only a biochemical pregnancy. According to the analysis combined with previous reports, there was no difference in the fertilization rate and pregnancy outcome parameters between TESE-ICSI and EJ-ICSI. Conclusions: TESE-ICSI for PCD may improve the fertilization rate compared to EJ-ICSI. However, it does not necessarily improve the pregnancy outcome for a patient with primary ciliary dyskinesia.