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Pseudo-Meigs' syndrome associated with malignant struma ovarii: a case report.

BACKGROUND: Malignant struma ovarii is a rare tumor, consisting of a struma ovarii with malignant transformation. The association of a malignant struma ovarii with pseudo-Meigs' syndrome and elevated Ca-125 levels has been never reported. CASE: A 66-year-old woman presented with monolateral ovarian mass, ascites, hydrothorax, and elevated Ca-125 levels. Optimal medical staging was performed. Definitive histological examination revealed a malignant struma ovarii. The immediate and complete resolution of symptoms and rapid decline of both Ca-125 and thyroglobulin levels to normal value were achieved post-operatively. After counseling, strict follow-up was planned, and no adjuvant therapy was administered. CONCLUSION: We report the first case of pseudo-Meigs' syndrome associated with malignant struma ovarii and elevated Ca-125 levels. The choice of not performing adjuvant therapy is feasible after optimal surgery and adequate staging procedure given to the usually clinical benign course and the low incidence of metastases in malignant struma ovarii. Careful patient counseling is required.

Aged↗

Pseudo-Meigs syndrome and elevated CA125 associated with struma ovarii.

BACKGROUND: Struma ovarii is a rare form of ovarian neoplasm composed entirely and predominantly of thyroid tissue. The association of pseudo-Meigs syndrome, elevation of CA125 and hyperthyroidism to struma ovarii is a rare condition. CASE: We report an unusual presentation of a postmenopausal woman with benign struma ovarii associated with pseudo-Meigs syndrome, hypertiroidism, and elevated CA125 serum level, and a large complex right pelvic mass thereby mimicking an ovarian cancer. CONCLUSIONS: Struma ovarii is a rare cause of ascites, hydrothorax, elevated CA125, and hyperthyroidism. This rare condition should be considered in the differential diagnosis in patients with ascites and pleural effusions but with negative cytology.

Aged↗

[Recurrence of Demons-Meigs' syndrome. A case report].

Meigs' syndrome includes ascites, hydrothorax and benign ovarian tumor. The aim of this study was to report a recurrence of Meigs' syndrome in a 71 year old woman, which occurred 30 years after the initial operation. It seems to be the first reported case of recurrence. The high level of CA125 was suggestive of malignancy. Resection of a peritoneal recurrence of the benign fibrothecoma was followed for the second time by improvement of all the symptoms.

Aged↗

Fetal lung lesions: management and outcome.

OBJECTIVE: Our purpose was to review our experience with fetal congenital cystic adenomatoid malformation and extralobar pulmonary sequestration emphasizing natural history, management, and outcome. STUDY DESIGN: We conducted a retrospective review of 175 fetal lung lesions diagnosed by antenatal ultrasonography at 2 fetal treatment centers. RESULTS: There were 134 congenital cystic adenomatoid malformation cases. Fourteen women underwent elective abortion, 101 women were managed expectantly, 13 women had fetal surgery, and 6 women had placement of a thoracoamniotic shunt. For the congenital cystic adenomatoid malformation lesions that were not associated with nonimmune hydrops, all babies survived. Of 25 large congenital cystic adenomatoid malformations that had associated hydrops that were followed expectantly, all fetuses died before or shortly after birth. Fetal surgical resection of the tumor (fetal lobectomy) was performed at 21 to 29 weeks' gestation in 13 hydropic fetuses with 8 fetuses continuing gestation with subsequent hydrops resolution, impressive in utero lung growth, and neonatal survival. Six fetuses with a very large solitary cyst underwent thoracoamniotic shunting and 5 survived. There were 41 extralobar pulmonary sequestration cases. Twenty-eight extralobar pulmonary sequestrations dramatically regressed on serial prenatal sonography, were asymptomatic after birth, and were only detectable by imaging studies postnatally (no resection required). Of the remaining 13 extralobar pulmonary sequestration cases, 2 underwent elective abortion, 7 symptomatic lesions were resected after birth with survival, 1 hydropic fetus died, and 3 fetuses had an associated tension hydrothorax with secondary hydrops that was successfully treated by either fetal thoracenteses or thoracoamniotic shunting followed by postnatal resection. CONCLUSIONS: The natural history of prenatally diagnosed lung masses is variable, and associated anomalies are rare. Most congenital cystic adenomatoid malformation lesions can be managed with maternal transport, planned term delivery, and postnatal resection. Many extralobar pulmonary sequestrations dramatically decrease in size before birth and may not need treatment after birth. Fetal therapy is now an option for lung lesions associated with nonimmune hydrops.

Amnion↗

Pleurovenous shunting in the treatment of nonmalignant pleural effusion.

BACKGROUND: The goals of treatment of chronic nonmalignant pleural effusion are relief of dyspnea and improved quality of life. Treatment options include needle thoracentesis, tube thoracostomy chemical pleurodesis, and pleurectomy. Pleurovenous shunting (PVS) represents an alternative, minimally invasive method. METHODS: Since 1999, 12 patients underwent pleurovenous shunting for right-sided pleural effusion in our center. Indications were hepatic hydrothorax (n = 6, one as bridging to liver transplantation), nephrotic syndrome (n = 4), and chylothorax (n = 2, one as bridging to lung transplantation). All patients received Denver shunt systems from the pleural cavity to either the subclavian or jugular vein. RESULTS: Shunt occlusion was observed in one case (chylothorax) 4 weeks after implantation. There was one early death, which was not related to the procedure (hepatic failure). No air embolism or infection was observed. All systems were patent throughout the observation period of 1 to 40 months (mean = 13.3 months), and none of the patients required further treatment for pleural effusion. CONCLUSION: Pleurovenous shunting offers an efficient, minimally invasive alternative to other surgical methods for treatment of recurrent nonmalignant pleural effusion.

Aged↗

Endocardial fibroelastosis in common domestic cats in the UK.

Endocardial fibroelastosis (EFE) is a pathological disorder characterized by a diffuse and elastic thickening of the endocardium. It is often thought to be congenital as most victims are infants who die of congestive heart failure before reaching one year of age. A naturally occurring animal model of EFE was proposed more than a decade ago on the basis of one pair of adult pure-bred Burmese cats which produced offspring consistently affected with EFE. In the present study a case of EFE is described which occurred in a closed colony of experimental SPF cats. A 5-week-old kitten which was found dead showed hydrothorax, hydropericardium and oedematous lungs at necropsy. Histological examination of the heart revealed left atrio-ventricular dilation with severe, diffuse endocardial thickening which did not extend into the myocardium. A retrospective study was carried out on stored tissues of two further kittens and one adult male cat from the colony which had died during a period of 2 years. The hearts of all the animals showed endocardial thickening in the left atria and ventricle but not in the right chambers. Over a period of 12 years of existence of this colony considerable inbreeding had occurred and it is concluded that an inherited factor was responsible for the disorder.

Animals↗

Systemic infection by equid herpesvirus-1 in a Grevy's zebra stallion (Equus grevyi) with particular reference to genital pathology.

A severe multi-systemic form of equid herpesvirus-1 infection is described in an adult zebra stallion. There was multifocal necrotizing rhinitis, marked hydrothorax and pulmonary oedema, with viral antigen expression in degenerating epithelial cells, local endothelial cells and intravascular leucocytes of the nasal mucosa and lung. Specific localization of EHV-1 infection was seen in the testes and epididymides, including infection of Leydig cells and germinal epithelium, which would have facilitated venereal shedding of virus in life. The case provided a unique opportunity to study hitherto undescribed aspects of the pathogenesis of naturally occurring EHV-1 infection in the male equine genital tract. Restriction digests of the isolate demonstrated a pattern similar to that of EHV-1 isolates previously recovered from aborted zebra and onager fetuses.

Animals↗

Prenatal diagnosis and the pediatric surgeon: the impact of prenatal consultation on perinatal management.

PURPOSE: Pediatric surgeons are increasingly called on by obstetrical colleagues to counsel parents about the implications of a prenatal ultrasound finding. Our understanding of the natural history of many prenatally diagnosed surgical conditions has grown significantly in recent years. Whether prenatal surgical consultation can influence perinatal course had not been investigated. METHODS: During an 21-month period, 12,865 prenatal ultrasound studies were performed on a total of 4,551 patients, and 221 prenatal surgical consultations were obtained through a newly established fetal treatment program at a tertiary care prenatal diagnostic center. To evaluate the impact of prenatal pediatric surgical consultation on perinatal course, the authors reviewed changes in management including termination of pregnancy, in utero intervention, and altered site, mode, or timing of delivery. RESULTS: Two hundred twenty-one fetuses were referred for consultation; their 234 congenital anomalies included genitourinary (36%), thoracic (16%), intraabdominal (14.5%), abdominal wall (10.6%), neurological (9%), skeletal (6%), and head and neck (2.5%) defects; 2.5% had tumors and 2.5% were twin pregnancies. Pregnancy was terminated in 9.5% of cases, because of patient request, chromosomal abnormality, or dismal prognosis. In 3.6%, the decision to terminate was changed as a result of consultation. Site of delivery was changed as a result of consultation in 37% to facilitate postnatal evaluation and initiate immediate treatment. Mode of delivery was changed in 6.8% to prevent dystocia, hemorrhage into a tumor, as in sacrococcygeal teratoma, or to provide an emergency airway, as in cervical teratoma. The timing of delivery was changed in 4.5% to avoid further damage to fetal organs in cases of obstructive uropathy, gastroschisis, sacrococcygeal teratoma with high-output failure, and hydrocephalus. Five percent (11) underwent treatment in utero for fetal hydrothorax, obstructive uropathy, twin-twin transfusion syndrome, or lymphangioma. The overall perinatal mortality rate was 2.5%. CONCLUSION: Prenatal pediatric surgical consultation may have a significant impact on the perinatal management of the fetus with a surgically correctable congenital anomaly. Providing obstetric colleagues and families with valuable insight into the surgical management of anomalies allows fetal intervention when appropriate, and delivery in an appropriate setting, by the safest mode of delivery, and at the gestational age appropriate to minimize effects of the anomaly.

Abortion, Therapeutic↗

Pulmonary sequestrations: prenatal ultrasound diagnosis, treatment, and outcome.

BACKGROUND/PURPOSE: With the development of antenatal diagnosis of pulmonary sequestrations, the authors decided to define more accurate perinatal operative indications. METHODS/RESULTS: Antenatal ultrasound scanning (US) enabled the diagnosis of congenital pulmonary malformation in 10 cases between the twentieth and the thirty-third week of amenorrhea (WA; average, 26 WA). An absolute or relative regression of the thoracic mass size was observed in five patients. The systemic arterial blood supply was identified in four patients by Doppler US. Two fetuses required treatment. One of them suffered from a voluminous sequestration, larger than one hemithorax, with polyhydramnios. Three successive paracentesis of ascites and amniotic fluid allowed the pregnancy to continue until term. The second fetus had a sudden left hydrothorax at 30 WA and was treated by a pleuroamniotic shunt. Five spontaneous partial involutions of the mass during the antenatal period were observed. The 10 patients underwent surgery after birth. There was no mortality. Morbidity occurred in one case of antenatal treatment. Twenty-eight other cases of antenatal diagnosis of pulmonary sequestration have been described in the medical literature. Spontaneous involution of the mass has been reported in eight fetuses and its complete disappearance in two cases. Thirteen fetuses had polyhydramnios. Five of these progressed spontaneously without treatment; only two survived. Two other fetuses were drained or punctured, and one survived. Premature deliveries were undertaken for the six other fetus; there was one perinatal death. CONCLUSIONS: Sequestrations with polyhydramnios may be treated in an early prenatal period. Mortality and morbidity rates are still high. At birth, large-sized sequestrations (more than half a hemithorax) must be operated on, even in cases of no respiratory distress. Medium-sized sequestrations must be operated on to remove the mass. Small and asymptomatic sequestrations must be operated on in case of intralobar forms (often cystic), or with a big blood supply. The artery may be responsible for severe complications (hemoptysis, aneurysm).

Bronchopulmonary Sequestration↗

Simultaneous bilateral percutaneous nephrolithotomy.

PURPOSE: We evaluate the results of simultaneous bilateral percutaneous nephrolithotomy. MATERIALS AND METHODS: The charts of 52 patients scheduled for simultaneous bilateral percutaneous nephrolithotomy at the Methodist Hospital of Indiana were retrospectively reviewed. The results of the 48 patients who underwent the procedure were tabulated and analyzed. RESULTS: Mean operative time was 269 minutes and mean hospital stay was 5.6 days. Of the patients 45 were rendered stone-free (96.9% of 96 renal units) and 3 had insignificant debris (fragments less than 4 mm.) unilaterally. Complications were infrequent and included hydrothorax in 5 cases, ureteral obstruction by fragment migration in 2, hematuria causing clot retention in 2, blood transfusion in 2 and ureteral perforation with a guide wire in 1. CONCLUSIONS: Simultaneous bilateral percutaneous nephrolithotomy is a well tolerated, safe, cost-effective and expeditious approach to patients with bilateral renal calculi requiring percutaneous nephrolithotomy.

Adolescent↗

The supracostal approach for percutaneous nephrostolithotomy.

Sixty-nine patients underwent percutaneous nephrostolithotomy or ultrasonic lithotripsy through a nephrostomy tract placed over the 12th rib. All procedures were accomplished as a single stage in a standard operating room with patients under general anesthesia. With the exception of a single failed access the targeted calculus was removed in all instances. There were no episodes of postoperative pneumothorax or hydrothorax. One patient suffered delayed postoperative pneumonia and another patient had a delayed pleural effusion. The supracostal approach for percutaneous nephrostolithotomy is an important and safe modification of the traditional percutaneous approach to the kidney for an endourological procedure.

Anesthesia, General↗

Percutaneous nephrolithotomy in the spinal cord injury population.

We evaluated 23 male spinal cord injury patients who underwent percutaneous nephrolithotomy for the success rate of stone removal and the incidence of operative complications. There were 18 quadriplegic and 5 paraplegic patients, and 5 had bilateral procedures. Of the kidneys 7 had staghorn calculi, 8 had pelvic and caliceal combinations, 6 had large multiple caliceal stones, 4 had large (more than 2.5 cm.) pelvic stones and 3 had pelvic stones less than 2.5 cm. Placement of a nephrostomy tube and stone extraction were performed as a single procedure with the use of general anesthesia in all but 4 patients. Our results showed that 19 of 21 compliant patients (90.4 per cent) were free of stone, with an average of 2.04 procedures per patient. A total of 47 procedures was performed, with an average operative time of 1 hour 45 minutes. Major complications were associated with 4 of the 47 procedures (8.5 per cent), and consisted of a respiratory arrest, 2 perirenal abscesses and a hydrothorax. Minor complications included fever (more than 101.5F) in 64.3 per cent and retained stones in 14.3 per cent of the kidneys operated upon, dislodged nephrostomy tubes in 12.6 per cent of the procedures (21.4 per cent of the kidneys operated upon), and anemia requiring transfusion in 17.0 per cent (8 of 47) of the procedures (27.8 per cent of the kidneys operated upon). The presence of infected stones, prior operative procedures and medical complexity of these patients make complications more frequent. Nevertheless, percutaneous nephrolithotomy is a safe and effective procedure for the spinal cord injury population.

Adult↗

Complications of chronic pancreatitis.

The most common complication of chronic pancreatitis is pain, which in many cases seems related to pancreatic ductal obstruction with ductal hypertension. Longitudinal pancreaticojejunostomy is indicated in patients with a dilated (larger than 7 mm) duct and pain that requires narcotic analgesics for relief. Chronic pseudocysts may be corrected surgically without the usual 6-week wait, and asymptomatic pseudocysts less than 4 cm in diameter may not require surgery at all. The relative efficacy and risks of percutaneous drainage of pseudocysts versus the standard surgical approaches need to be studied. Pancreatic fistulas may be external or internal, where pancreatic ascites or hydrothorax can be the clinical manifestation. The pharmacologic suppression of pancreatic secretion (e.g., with somatostatin) may be useful in their management, but surgery may be required. Pancreatic resection or internal drainage is usually effective. Persistent jaundice should be relieved surgically by choledochoduodenostomy to avoid the development of secondary biliary cirrhosis. Obstruction at various levels of the gastrointestinal tract (duodenum, small bowel, colon) may require bypass (gastrojejunostomy) or resection. Hemorrhage from major arteries is an infrequent but often lethal complication of chronic pancreatitis, especially associated with pseudocysts. Angiography is invaluable for diagnosis and occasionally for treatment (embolization). Surgery is preferred in good-risk patients, with suture ligation (resection) of the bleeding source. Chronic pancreatitis is the most common cause of splenic vein thrombosis. The resultant hemorrhage from gastric varices is managed effectively by splenectomy.

Chronic Disease↗

Urologist-acquired renal access for percutaneous renal surgery.

OBJECTIVES: In most endourology programs an interventional radiologist is employed to acquire renal access for percutaneous renal surgery. Over the last 13 years the senior endourologist at Oregon Health Sciences University has acquired access without employing a radiologist. We report our experience with urologist-acquired renal access for percutaneous renal surgery in 522 cases. METHODS: We reviewed the records of all patients at our hospital who underwent percutaneous renal surgery between August 1983 and December 1996 with renal access being obtained in the operating room by a urologist. RESULTS: Four hundred fifty-six patients underwent 522 procedures. Indications for percutaneous renal surgery were renal and proximal ureteral calculi (n = 516), retained ureteral stent (n = 3), and intrarenal collecting system tumor (n = 3). We were successful in gaining access to 513 of 522 kidneys (98.3%). Access was obtained via a subcostal approach in 344 procedures, over the 12th rib in 152 procedures, over the 11th rib in 15 procedures, and transabdominally in 2 procedures. Sixty-five patients (12.7%) required a second or multiple sites to facilitate complete removal of calculi. Our overall complication rate was 15.3%. Blood transfusion was required in 5.4% of the cases, ileus developed in 1.9%, pneumothorax in 1.1%, intraoperative hydrothorax in 1.1%, postoperative pleural effusion requiring aspiration in 0.9%, and septic shock in 0.9%. Our overall success rate for stone removal was 94.5%. CONCLUSIONS: In our experience, the urologist is able to safely and effectively obtain percutaneous access to the collecting system for percutaneous renal surgery as a one-stage procedure without the aid of interventional radiologists.

Adolescent↗

Meigs' syndrome in a young woman.

Meigs' syndrome is a rare entity in women under age 30 years. It is characterized by a benign fibrous ovarian tumor, ascites, and hydrothorax. Complete resolution of symptoms occurs with removal of the tumor. The case of a 20-year-old woman with a fibroma and dermoid cyst is presented and the literature reviewed. The etiology of the fluid accumulations remains unclear, although it appears to be related to lymphatic obstruction. Presenting symptoms may include abdominal distension and pain, menstrual irregularities, cough, pleuritic chest pain, and weight gain. Meigs' syndrome and pseudo-Meigs' syndrome should be included in the different diagnosis in young women presenting with a pleural effusion. Resolution of the fluid accumulation usually occurs within 2 weeks of tumor removal.

Adult↗

Ten years' experience with continuous ambulatory peritoneal dialysis.

Up to January 1989, 171 patients were trained at our center on continuous ambulatory peritoneal dialysis (CAPD), and 17 on continuous cyclic peritoneal dialysis (CCPD). Over 10 years, we have gained 5,068 patient-months experience. Patient survival was 60% and 31% at 5 and 10 years, respectively. In contrast, diabetics had a survival of 32% at 5 years. Major complications included 499 new episodes of peritonitis, 304 exit-site infections, 22 hernias, five bowel perforations, one hydrothorax, and three episodes of sclerosing encapsulating peritonitis. Our technique survival has been 62% and 40% at 5 and 10 years, respectively. We believe that CAPD is a viable dialysis technique for long-term treatment of chronic renal failure and it should be offered as an option to intermittent hemodialysis.

Adolescent↗

Meigs' syndrome with elevated CA125: case report and review of the literature.

A 51-year-old woman presented with acute ascites and hydrothorax is reported. Pelvic ultrasound showed two pelvic solid masses and serum CA125 level was elevated (577 IU/ml). Pathology revealed bilateral ovarian fibromas. We present this case of Meigs' syndrome and discuss its diagnostic problems and a review of the literature.

Abdomen↗

[Traumatic diaphragmatic rupture with delayed unusual disclosure].

The authors report the case of a delayed presentation of a traumatic diaphragmatic rupture in a 22-year-old patient admitted to hospital for a minor surgical procedure under general anaesthesia. Nine months before, he had a road traffic accident with a minor thoracic trauma. Three days after surgery, the patient was readmitted for a tension hydrothorax due to the herniation and the perforation of the stomach into the left pleural cavity. Such a delayed presentation of a traumatic diaphragmatic rupture remains uncommon. The peroperative ventilatory factors involved in the development of the hernia are discussed.

Accidents, Traffic↗