[Solitary genital leiomyoma of the tunica dartos: a case report and review of the literature in Japan].

We report a case of solitary genital leiomyoma arising from the tunica dartos of the scrotum. A 43-year-old man complained of a slightly tender left scrotal mass that had enlarged over approximately 17 years. On physical examination, the tumor was pedunculate and attached to the lower pole of the left scrotum. The tumor was clearly separate from scrotal contents such as the testis, epididymis, and spermatic cord. Ultrasonography showed the tumor to be 20.0 X 23.5 mm in diameter. Other laboratory examination findings were normal. Under local anesthesia, the tumor was resected at its base with overlying skin. On histopathological examination, the tumor consisted of interlacing bundles of smooth muscle cells, which were positive for vimentin, desmin, and a-actin. The tumor was diagnosed as leiomyoma arising from the scrotal tunica dartos. As solitary genital leiomyoma of the tunica dartos is rare, we have presented this case history and reviewed 23 cases that occurred in Japan.

[Postoperative hypoglycemia after resection of pheochromocytoma: a case report].

We report a case of severe hypoglycemia following resection of pheochromocytoma. A 39-year-old male was admitted to our hospital with a chief complaint of palpitation. Blood pressure and fasting blood glucose were within the normal range. Computed tomography and magnetic resonance imaging revealed a right adrenal tumor (7.5 x 5 x 7 cm) and 131I-MIBG scintigraphy showed marked tumor uptake of isotope. As plasma and urinary catecholamine levels were very high, a clinical diagnosis of pheochromocytoma was established. Doxazosin and propranolol were administered for 43 days prior to adrenalectomy. During intensive care monitoring 2 hours postoperatively, the patient became drowsy and began to sweat. Although blood pressure remained stable, severe hypoglycemia (38 mg/dl) and hyperinsulinism (63.67 microU/ml) were confirmed. Infusion of 50% glucose improved these symptoms and plasma glucose level and insulin secretion normalized within 15 days of surgery. We also reviewed 25 cases of hypoglycemia after resection of pheochromocytoma. We recommend close monitoring of blood glucose for at least 6 hours after adrenalectomy for pheochromocytoma.

[A case of late recurrence of germ cell tumor].

A 19-year-old male was admitted to our hospital because of retroperitoneal tumor and metastases in the lung and liver. He underwent chemotherapy followed by retroperitoneal lymph node dissection and pathologic examination revealed mostly necrotic tissue with a small amount of teratomatous tissue. Two years later, a hyper echoic lesion was found in the left testis, and left high orchiectomy was performed. The resected specimen appeared to be a burned-out testicular tumor. Ten years later, CT scan detected lymph node swelling in the retroperitoneum, and excision of the tumor was performed. Pathologic examination revealed well-differentiated adenocarcinoma. Since no primary adenocarcinoma was found, this case was considered late relapse of a germ cell tumor.

Comparative study of novel endoluminal ultrasonography and conventional transurethral ultrasonography in staging of bladder cancer.

BACKGROUND: Recent advances in ultrasonic techniques have improved the image quality and diagnostic accuracy for staging of bladder cancer. The aim of this study was to assess the feasibility and usefulness of endoluminal ultrasonography (ELUS) in staging of bladder cancer, and to compare them with those of conventional transurethral ultrasonography (TUUS). METHODS: From 2000 to 2002, 19 patients with bladder cancer were evaluated by ELUS and TUUS before transurethral resection or biopsy. Clinical staging using ELUS, TUUS, computed tomography (CT) and magnetic resonance imaging (MRI) was compared with the results of pathological staging. RESULTS: In 16 of 19 patients, both ELUS and TUUS were able to diagnose tumor stage. In the remaining three patients, both methods were unable to evaluate stage of tumor. In two of these patients, this inability to evaluate tumor state was caused by a difficulty in depicting the tumor base in rectangular scanning. In the remaining patient, the inability to evaluate tumor stage was caused by a difficulty in recognizing the normal muscularis because of edema around the tumor base. Both diagnostic accuracies of ELUS and TUUS were 84%, which were superior to those of CT (44%) and MRI (82%). CONCLUSIONS: Endoluminal ultrasonography and TUUS were equally useful for staging diagnosis of bladder cancer. Because the ELUS probe is very small in diameter and can be manipulated under direct vision, it is superior to the TUUS in safety and in fine visualization. However, the main limitations of ELUS include an inability to evaluate the depth of invasion of large tumors and an inability to visualize the tumor base in the position of the bladder neck.

[Paravesical granuloma after inguinal herniorrhaphy].

Paravesical granuloma is a rare complication after inguinal herniorrhaphy. We report a case of this rare disease and review 27 previously reported cases. A 70-year-old male presented with hematopyuria. He had undergone right inguinal herniorrhaphy five years earlier. On presentation, the right inguinal area was wet with exudate. Cystoscopy revealed edematous mucosa on the right side of the bladder dome, but transurethral bladder biopsy demonstrated no malignancy. CT identified a 2-cm diameter mass with heterogeneous appearance on the right side of the bladder dome. Treatment with antibiotics proved ineffective and en bloc excision of the tumor with partial cystectomy was performed. Symptoms subsequently resolved completely. Histopathologically, the tumor represented inflammatory granuloma, and a mesh thought to be a remnant from the previous herniorrhaphy was found in the central portion of the tumor. Paravesical granuloma should be considered for patients with continuous hematopyuria and a history of inguinal herniorrhaphy.

Reversal of acquired cisplatin resistance by modulation of metallothionein in transplanted murine tumors.

BACKGROUND: The platinum-based chemotherapeutic agent cisplatin is involved in a broad spectrum of activities against human systemic malignancies. However, acquired resistance to cisplatin reduces its clinical efficacy. Elucidation of the molecular basis of cisplatin resistance is required to improve the effectiveness of cisplatin. In the present study, the mechanism of acquired resistance to cisplatin was studied in C3H mice inoculated with MBT-2 murine bladder tumor cells. METHODS: C3H mice were subcutaneously inoculated with 1.0 x 10(6) MBT-2 cells/mouse on day 0. The mice were given intraperitoneal injections of 10 micro mol/kg cisplatin and subcutaneous injections of 1000 micro mol/kg propargylglycine, an inhibitor of gamma-cystathionase, once a day for 10 consecutive days from day 11 to day 20. RESULTS: The metallothionein content of the tumors was increased to twice the control level by repeated administration of cisplatin. Co-administration of propargylglycine reduced metallothionein induction in the tumors and markedly enhanced the antitumor activity of cisplatin. In contrast, the glutathione content in the tumors did not change from the control level after cisplatin administration. The platinum accumulation in tumors treated with cisplatin alone was 1.7-fold greater than when propargylglycine was administered concomitantly. The platinum concentrations changed in accordance with the metallothionein contents. CONCLUSIONS: These observations suggest that metallothionein, but not glutathione or reduced platinum accumulation, might play a role in the acquired resistance to cisplatin of C3H mice inoculated with MBT-2. Moreover, reversal of this resistance might be possible by biochemical modulation of metallothionein.

[Ammonium acid urate stone due to laxative abuse: a case report].

We report a case of ammonium acid urate stone due to laxative abuse. A 27-year-old female complained of left flank pain. Computed tomography revealed bilateral ureter stones (right 16.5 x 9.0 mm; left 4 mm), while left ureter stone was radiolucent on the plain X ray film. Bilateral hydronephrosis was seen, but no therapy was performed for the right stone, because 99mTc-MAG3 scintigraphy revealed that right kidney had no function. The left stone was successfully removed by transurethral approach. The stone was revealed to be an ammonium acid urate by infrared spectrophotometry. She had been taking many laxatives (bisacodyl, sennoside, aloe extract) for 12 years to control her body weight. Ammonium acid urate stones are rarely seen in developed countries. We have reviewed 9 cases in Japan, describing ammonium acid urate stones due to laxative abuse. Among these patients, 24-hour urine volume and excretion in urinary sodium were decreased, and serum aldosterone was increased. The involvement of laxative abuse should be considered when ammonium acid urate is formed in a woman with a low body mass index.

[Tumor enucleation for pelvic schwannoma: a case report].

A 36-year-old man with a complaint of pollakisuria visited our hospital. A non-displaceable, palm-sized tumor was palpable in the lower abdomen. Laboratory data were normal except for slightly high serum S100 protein. Intravenous pyelography revealed hydronephrosis on the right side and deviation of the bladder to the left. Computed tomogaphy revealed a heterogenous tumor in the pelvis with a cystic lesion and calcification. The tumor was 16 x 12 x 11 cm in size and in contact with the sacrum. The tumor was extirpated following diagnosis as a benign schwannoma by needle biopsy. The pelvic cavity was occupied by the tumor rigidly adhered to the sacrum. Although serious complications, such as bleeding and nerve injury were apprehended, we incised the tumor capsule and enucleated the contents as much as possible. The histopathological diagnosis of the resected specimen was benign schwannoma, type Antoni A. In the last 10 years, 37 cases of pelvic schwannoma have been reported in the Japanese literature. In most of them, surgical extirpation was difficult to perform because of adhesion to the sacrum. If the tumor is confirmed benign from histopathologic findings preoperatively, tumor enucleation may become a therapeutic option.

[Pulmonary rhabdomyosarcoma generated during treatment of testicular tumor].

A 23-year-old male was admitted to our hospital for the management of pulmonary metastases. He had undergone right high orchiectomy, chemotherapy with four courses of PEB regimen (cisplatin, etoposide, bleomycin) and retroperitoneal lymph node dissection the previous year. The pathological findings showed mixed germ cell tumor (seminoma, yolk sac tumor, embryonal carcinoma) in the testis and mature teratoma in the draining lymph node. Two courses of salvage chemotherapy using a VIP regimen (etoposide, ifosfamide, cisplatin) were performed after diagnosis of pulmonary metastases, but had no affect on tumor size. Video-assisted excision of pulmonary metastases was then performed, giving a pathological diagnosis of rhabdomyosarcoma in all three resected tumors. The operation was followed by three courses of CYVADIC (cyclophosphamide, vincristine, adriamycin, dacarbazin) chemotherapy and oral cyclophosphamide, as a small residual tumor was suspected. These chemotherapeutic interventions have appeared effective, with no apparent recurrence of lesions at present, one year after the excision of pulmonary metastases.

[A case of perineal abscess due to urethral fistula in a patient with spinal cord injury].

We report a case of perineal subcutaneous abscess due to urethral fistula in a patient with spinal cord injury. A 39-year-old male visited our hospital complaining of left scrotal swelling and fever. The left scrotum and perineal skin were swollen to the size of a goose egg, and pus was discharged from the perineal swollen bump. Magnetic resonance imaging (MRI) suggested an urethral fistula with a large subcutaneous abscess. The abscess was resected with debridement of necrotic tissue, and a cystostomy was placed. Endoscopy revealed a fistula in the bulbar urethra. The characteristics of this rare entity are discussed.

[A case of penile malignant melanoma].

A 79-year-old male presented in December. In January, 2001, with complaints of black nodules and bleeding from the glans of the penis to the foreskin. Inguinal lymph nodes were palpable bilaterally. Clinical diagnosis was penile malignant melanoma. Cystoscopy and urethrography revealed urethral invasion of malignant melanoma, and magnetic resonance imaging (MRI) of the penis revealed invasion to prostate, and pelvic lymph node metastases in abdominal compuled tomography (CT) but no organ metastases. Total cystectomy, total penectomy, bilateral inguinal and pelvic lymph node dissection and bilateral ureterocutaneostomy were performed in February, 2002. The pathological findings were nodular malignant melanoma, pT4bN2bM1a, and the surgical margin was positive. After these therapies, chemotherapy was performed. Five months later, CT revealed multiple lung and brain metastases, and radiation therapy and chemotherapy were performed. Twelve months after the operation, he died of cancer. Review of the literature revealed that our patient is the thirtieth reported case of penile malignant melanoma in Japan since 1924. In 30 cases, stage III, IV were 20 cases and 16 cases performed operation.

[Two cases of vesical nephrogenic adenoma].

Case 1: A 52-year-old man receiving regular treatment for quadriplegia due to Friedreich disease visited our hospital with the chief complaint of macroscopic hematuria. He had undergone cystostomy 12 years ago due to neurogenic bladder. The computed tomography and cystoscopic examination revealed a bladder tumor with a few bladder stones. Transurethral resection of bladder tumor (TUR-Bt) was performed after bladder stone removal in May 2000. The pathological diagnosis showed nephrogenic adenoma. Case 2: A 54-year-old man had been treated with bladder tumor by TUR-Bt in Nov. 1995. The pathological diagnosis showed transitional cell carcinoma, G3, pT2 and intravesical instillation therapy using THP was performed. The bladder tumor had recurred twice and the instillation therapy had been exchanged to BCG since Nov. 1997. A small bladder tumor was observed in Jan. 2001, and from the biopsy specimen it was diagnosed as nephrogenic adenoma. Forty-six cases of urothelial nephrogenic adenoma including our cases have been reported in Japan. Chronic stimulation such as bladder stone and infection is thought to induce nephrogenic adenoma. BCG instillation therapy is believed to be an initiation factors for nephrogenic adenoma.

[Retroperitoneal germ cell tumor with testicular calcification indicating tiny testicular origin: consideration of the origin of retroperitoneal germ cell tumors: report of two cases].

Two cases of germ cell neoplasm retrospectively considered to have been of testicular origin are reported. Case 1. A 19-year-old male with brain, liver and retroperitoneal tumors was diagnosed with yolk sac tumor by retroperitoneal tumor biopsy. After multidisciplinary treatment, a region of calcification was detected in the left testis on scrotal sonography and left high inguinal orchiectomy was performed. Case 2. A 57-year-old male with neck, lung and retroperitoneal tumors was diagnosed with yolk sac tumor by supraclavicular biopsy. From initial examination, scrotal sonography revealed a small calcified lesion in the right testis. After chemotherapy, high inguinal orchiectomy and retroperitoneal lymphadenectomy were simultaneously performed. Pathologic evaluation of these testicular specimens revealed calcification and a fibrous scar in correspondence with the clinical diagnosis. These changes were considered as scars of the primary testicular tumor due to burned-out tumor or the result of reaction to chemotherapy. Since a primary tumor of testicular origin may exist in the extragonadal germ cell tumor, it is important to examine the intrascrotal contents in detail in the case of so-called extragonadal germ cell tumors with palpably normal testes. In such cases, there are two possible conditions, an occult testicular tumor and a burned-out testicular tumor. We briefly reviewed 42 such cases in the Japanese literature. It appears that there are very few true extragonadal germ cell tumors, and that the possibility of primary testicular origin metastasizing from viable occult testicular tumor or burned-out testicular tumor with spontaneous regression is high in retroperitoneal germ cell tumors.

[Long-term study of tiopronin in patients with cystinuria].

Thirty two patients with cystinuria were enrolled in this long-term study and 16 patients were treated with tiopronin for 24 weeks. Tiopronin reduced daily urinary cystine excretion from 901.48 mg (before treatment) to 488.60 mg (on the average of 12th week and 24th week after tiopronin administration) successfully. Tiopronin therapy was tolerated well, but side effects were observed in 13 events in 6 patients. Thus tiopronin was expected to be effective in preventing cystine stone formation and tolerated well.

[A case of erythropoietin-producing renal cell carcinoma with polycythemia].

The patient was a 46-year-old man with gross hematuria and left lumbar pain. Computed tomography revealed a large left renal tumor with hemorrhage. Serological examination revealed polycythemia and a high erythropoietin level. After left radical nephrectomy, polycythemia and serologically high level of erythropoietin disappeared. Histopathological findings showed renal cell carcinoma composed of spindle cells. Based on the above mentioned clinical course, we diagnosed this case as erythropoietin-producing renal cell carcinoma with polycythemia. It is suggested that erythropoietin not only causes polycythemia but also stimulates proliferation of the tumor, because the majority of erythropoietin-producing renal cell carcinomas have been reported to be highly advanced. This patient had no evidence of disease one year after the operation.

[A case of spermatic granuloma difficult to differentiate from malignant tumor].

A 33-year-old man visited our hospital with complaint of painless left intrascrotal mass. A hard and rough induration in contact with the left testis was palpable. The laboratory data including beta-human churionic gonadotropin, alpha fetoprotein and lactate dehydrogenase were normal. Ultrasound sonography showed a large hypoechoic lesion at the left epididymis. Surgical exploration of this lesion indicated a malignant tumor of the epididymis or spermatic cord, and left high orchiectomy was performed. A milk-white nodule 6 cm in diameter was found in the resected specimen. Pathological diagnosis was spermatic granuloma. Fourteen cases of spermatic granuloma have been reported in the last twenty years in Japan. All of them were relatively small nodules and epididymectomy was performed for most of them. We selected radical orchiectomy because of a large nodule with suspicion of malignant lesion.

Immunohistochemical expression of metallothionein in human bladder cancer: correlation with histopathological parameters and patient survival.

PURPOSE: We assessed metallothionein expression and its patterns of distribution as possible prognostic variables in bladder cancer with regard to histopathological parameters. MATERIALS AND METHODS: We stained 91 formalin fixed, paraffin embedded tissue specimens of bladder cancer from 91 patients with no history of treatment using an immunohistochemical technique for metallothionein. Relationships between immunoreactivity for metallothionein and histopathological parameters were examined. In addition, these parameters, including metallothionein, were evaluated as potential prognostic markers. RESULTS: Metallothionein was detected in 33 of 91 bladder cancers (36.3%). There were significant relationships of metallothionein expression with high grade, high stage and nonpapillary growth pattern tumors. There was no specific correlation of metallothionein expression with the interval to intravesical recurrence. In the 31 patients who underwent radical cystectomy a significant relationship between metallothionein immunoreactivity and cancer specific survival was found. CONCLUSIONS: Our findings suggest that a close correlation exists of metallothionein expression with histopathological parameters and metallothionein expression can be a useful prognostic variable for bladder cancer.

Increased expression of mRNAs for microtubule disassembly molecules during nerve regeneration.

The mRNA expression of the microtubule disassembly molecules (SCG10, stathmin, SCLIP and RB3) in response to nerve injury was examined using a rat hypoglossal nerve injury model. After nerve injury prominent increase in mRNA expression of SCG10, stathmin and RB3 was observed, while only slight increase in SCLIP mRNA was observed in injured motor neurons. The increase in SCG10 and RB3 mRNA expression was quicker than that of stathmin and SCLIP. All the elevated signals decreased gradually to control levels by 4 weeks after nerve injury.

[Nephrogenic adenoma of the bladder in children].

We report an unusual case of a 3-year-old girl with multiple nephrogenic adenomas in the urinary bladder following previous surgical intervention. When she was 6-month-old, right vesicoureter-alreflux (VUR) and left marked hydronephrosis with ectopic urethral opening were found. The left renal pelvis and ureter were incompletely duplicated with a short common segment. Bilateral ureterocystoneostomy and closure of ectopic opening of the left ureter were performed. Left ureteral orifice resulted in double barreled pattern. Postoperative voiding-cystourethrography revealed VUR in the ureter belonging to the lower part of the left kidney. At the age of 3 year, cystoscopic examination revealed multiple papillary lesions in the urinary bladder. These lesions were resected transurethrally and the pathologic diagnosis was nephrogenic adenoma. The patient is the 27th case of nephrogenic adenoma of bladder reported in the Japanese literature.

Relation between technetium 99m-methoxyisobutylisonitrile accumulation and multidrug resistance protein in the parathyroid glands.

Technetium 99m ((99m)Tc)-methoxyisobutylisonitrile (MIBI) scintigraphy has been reported to be a useful method for visualizing the parathyroid glands for the diagnosis of hyperparathyroidism. Some drug metabolism transporters, such as p-glycoprotein (P-GP) or multidrug resistance-associated protein (MRP), are believed to be involved with one mechanism of (99m)Tc-MIBI accumulation in the parathyroid glands. We analyzed the expression of P-GP and MRP in 40 parathyroid glands from eight patients with primary hyperparathyroidism and six patients with secondary hyperparathyroidism using an immunohistochemical procedure. These patients underwent (99m)Tc-MIBI scintigraphy and thallium 201 ((201)Tl)/(99m)Tc subtraction scintigraphy preoperatively. We investigated the relation between the scintigraphic images and the expression of P-GP and MRP. The positive findings of (99m)Tc-MIBI accumulation in the hyperfunctioning parathyroids were increased significantly. There was a significant difference between the expression of P-GP and the (99m)Tc-MIBI scintigraphy findings. High expression of P-GP in parathyroid cells resulted in the negative image of (99m)Tc-MIBI, and low or no expression of P-GP made the image positive. There was no significant difference between the expression of MRP and the (99m)Tc-MIBI scintigraphic imaging. The (99m)Tc-MIBI image was also related to the weight of the parathyroid glands but not to the type of the parathyroid cells. Based on these clinical findings, we can hypothesize that P-GP functions as a drug transporter not only for chemotherapeutic agents but also for (99m)Tc-MIBI.