Cases reported "Syndrome"

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1/517. An extrahepatic bile duct metastasis from a gallbladder cancer mimicking Mirizzi's syndrome.

    We report a case of an extrahepatic bile duct metastasis from a gallbladder cancer that mimicked Mirizzi's syndrome on cholangiography. A 67-yr-old woman was admitted to our hospital with a diagnosis of acute calculous cholecystitis. As obstructive jaundice developed after the admission, percutaneous transhepatic biliary drainage was performed to ameliorate the jaundice and to evaluate the biliary system. Tube cholangiography revealed bile duct obstruction at the hepatic hilus, and extrinsic compression of the lateral aspect of the common hepatic duct, with nonvisualization of the gallbladder. No impacted cystic duct stone was visualized on CT or ultrasonography. laparotomy revealed a gallbladder tumor as well as an extrahepatic bile duct tumor. We diagnosed that the latter was a metastasis from the gallbladder cancer, based on the histopathological features. This case is unique in that the extrahepatic bile duct metastasis obstructed both the common hepatic duct and the cystic duct, giving the appearance of Mirizzi's syndrome on cholangiography. Metastatic bile duct tumors that mimic Mirizzi's syndrome have not been previously reported. The presence of this condition should be suspected in patients with the cholangiographic features of Mirizzi's syndrome, when the CT or ultrasonographic findings fail to demonstrate an impacted cystic duct stone.
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2/517. cytomegalovirus associated neonatal pneumonia and Wilson-Mikity syndrome: a causal relationship?

    lung injury caused by intrauterine inflammation has recently been strongly implicated in the pathogenesis of Wilson-Mikity syndrome (WMS). This article supports this theory by suggesting a causative role of intrauterine cytomegalovirus (CMV) infection for the development of WMS. A male premature infant, born at 33 weeks of gestational age, developed chronic lung disease compatible with WMS and diagnostic evaluation was positive for CMV infection. High-resolution computed tomography scan and lung histology revealed typical features of WMS in association with signs of interstitial pneumonia. CMV was found in urine, breastmilk, bronchoalveolar lavage material and lung tissue from open lung biopsy. Follow-up after treatment with ganciclovir and steroids showed resolving lung disease at the age of 6, 10 and 16 months, with lung function signs of mild obstruction. Assuming that a chance coexistence of cytomegalovirus pneumonia and Wilson-Mikity syndrome is rather unlikely, it is possible that intrauterine cytomegalovirus infection caused a pattern of lung injury consistent with Wilson-Mikity syndrome. Further cases of Wilson-Mikity syndrome should be investigated as to a possible role of congenital infection.
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3/517. Functional intestinal obstruction due to deficiency of argyrophil neurones in the myenteric plexus. Familial syndrome presenting with short small bowel, malrotation, and pyloric hypertrophy.

    In 3 infants functional intestinal obstruction, associated with a short small intestine, malrotation, and pyloric hypertrophy, was shown to be due to failure of development of the argyrophil myenteric plexus, with the absence of ongoing peristalsis. 4 infants with similar clinical features have been described previously, and there is evidence for an autosomal recessive mode of inheritance of this syndrome.
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keywords = obstruction
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4/517. The Rapunzel syndrome (trichobezoar) causing atypical intussusception in a child: a case report.

    The Rapunzel syndrome (trichobezoar) occurs when gastrointestinal obstruction is produced by a rare manifestation of a trichobezoar with a long tail that extends to or beyond the ileocecal valve. A case history of a 7-year-old girl is described. This is the eleventh patient with Rapunzel syndrome reported in the literature. The epidemiology, clinical features, diagnosis, complications, treatment, and literature are reviewed briefly.
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5/517. Intestinal blind pouch- and blind loop- syndrome in children operated previously for congenital duodenal obstruction.

    A follow-up study of 27 children operated for congenital duodenal obstruction (CDO) in the years 1953--71 is presented. Nine children belonged to the intrinsic and 18 children to the extrinsic group of CDO. A total of 7 retrocolic, isoperistaltic, side-to-side duodeno-jejunostomy, 7 Ladd's operation, 8 duodenolysis, 2 reduction of midgut volvulus, 2 duodenostomy a.m. Morton and one gastro-jejunostomy were performed at the age of 1 day--15 years. The clinical and radiological examinations were performed 3--21 years (mean 10 years 2 months) after these operations. In 3 cases there was a moderate duodenal dilatation, but reoperation was not necessary. During the follow-up period, one boy, now aged 8 years, developed a blind pouch-syndrome in the I portion of the duodenum containing a 5 x 5 cm phytobezoar 4 1/2 years after duodeno-jejunostomy. The frequency of blind pouch-syndrome after duodeno-jejunostomy was thus 1:7 or 14%. One girl, now aged 9 years, developed a blind loop-syndrome in the ileocaecal segment 3 months after side-to-side ileotransversostomy, which was performed from adhesion-obstruction after duodenolysis for malrotation I and CDO. Both the blind pouch- and the blind loop-deformation were resected and the children recovered well. To avoid blind-pouch- and blind loop-deformations in the intestines, the anastomosis must be made wide enough, and especially in the surgery of the jejuno-ileo-colic region an end-to-end anastomosis is preferable.
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ranking = 6
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6/517. duodenal obstruction by gallstone: case report of Bouveret's syndrome.

    Bouveret's syndrome involves gastric outlet obstruction by gallstone. Herein we describe an unusual case of duodenal bulb obstruction by gallstone. An 80-year-old woman was hospitalized with a fifteen-day history of vomiting. Computed tomography (CT) showed pneumobilia and a round calcified mass in the second portion of the duodenum. upper gastrointestinal tract series demonstrated the same sized oval radiolucency between the bulbus and the second portion of the duodenum. Endoscopic examination revealed a round black mass in the second portion of the duodenum, totally occupying the lumen. Endoscopic removal and destruction of the gallstone was attempted using a dye-laser, but the stone was too hard to crush. Eventually surgical enterolithotomy was successfully performed without cholecystectomy or closure of the fistula. Improved preoperative systemic management and prompt examination allowed earlier surgical intervention and reduced the morbidity. Surgical approach whether fistula closure should be performed remains controversial.
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ranking = 6
keywords = obstruction
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7/517. Trousseau's syndrome with brachiocephalic vein thrombosis in a patient with uterine carcinosarcoma. A case report.

    The authors treated a patient with the previously unreported occurrence of brachiocephalic vein and superior vena cava thrombosis in association with a distantly located cancer. A 71-year-old woman presented with swelling over the right side of the neck and abdominal distension. physical examination revealed a huge mass, and computed tomography demonstrated thrombosis of the brachiocephalic vein and superior vena cava accompanied by jugular vein dilatation. No coagulation disorder was demonstrable. After anticoagulation and thrombolysis, hysterectomy was performed; microscopic examination of the specimen revealed uterine carcinosarcoma. Even though local tumor obstruction is a much more common cause of neck vein thrombosis, a distant occult cancer can present as this form of Trousseau's syndrome. In patients with otherwise unexplained neck vein thrombosis, examination not only of the head and neck but also of the abdomen and pelvis should be pursued.
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8/517. Moebius syndrome: the new finding of hypertrophy of the coronoid process.

    The first detailed description of congenital facial paralysis was reported by Moebius in 1888. It is characterized by either unilateral or bilateral paralysis of the facial muscles and an associated abducens palsy. The present report is of two patients with Moebius syndrome, who were also diagnosed with trismus at birth. Each patient also demonstrated bilateral hypertrophy of the coronoid process of the mandible. In effect, the zygoma obstructed the excursion of the mandible because of a "coronoid block." A three-dimensional computed tomography scan demonstrated normal temporomandibular joints but bilateral hypertrophy of the coronoid processes and micrognathia. Both patients demonstrated less than 10 mm of oral excursion. Bilateral coronoidectomies were performed through an intraoral approach. The oral excursions after surgery increased to at least 20 mm. In each of these patients, the coronoid process was enlarged relative to the zygoma, which was of normal size and configuration. The trismus was associated with blocking of the coronoid by the anterior zygoma, preventing open or full excursion of the hypoplastic mandibles. Moebius syndrome can have a variable presentation at birth. In two patients, the authors describe a new finding of hypertrophy of the coronoid process and trismus secondary to obstruction of the coronoid by the hypertrophic zygomas during oral excursions. Each patient is described, and a review of the literature is discussed.
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keywords = obstruction
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9/517. A malignant glaucoma-like syndrome following pars plana vitrectomy.

    OBJECTIVE: To report two cases of a malignant glaucoma-like syndrome following pars plana vitrectomy. DESIGN: Two interventional case reports. INTERVENTION: The first patient was treated with a neodymium:YAG laser peripheral iridectomy with hyaloidectomy and with intracameral tissue plasminogen activator. The second patient was treated with a posterior approach iridectomy through residual hyaloid, zonules, and iris. MAIN OUTCOME MEASURES: Axial anterior chamber depth and intraocular pressure (IOP). RESULTS: The interventions resulted in deepening of the anterior chambers and normalization of IOPs. CONCLUSION: A pseudomalignant glaucoma syndrome may be related to obstruction of aqueous flow, either by residual anterior hyaloid or by fibrin and other inflammatory debris at the level of the ciliary body-zonular apparatus. Treatment of this syndrome involves restoring aqueous flow to the anterior chamber by disrupting the residual anterior hyaloid or clearing fibrin or inflammatory debris. The clinician should not disregard the possibility of a pseudomalignant glaucoma syndrome following vitrectomy despite the fact that vitrectomy has traditionally been considered a curative treatment for malignant glaucoma.
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ranking = 1
keywords = obstruction
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10/517. Ocular anterior chamber dysgenesis in craniosynostosis syndromes with a fibroblast growth factor receptor 2 mutation.

    Fibroblast growth factor receptor (FGFR) mutations have been found in craniosynostosis syndromes with and without limb and/or dermatologic anomalies. Ocular manifestations of FGFR2 syndromes are reported to include shallow orbits, proptosis, strabismus, and hypertelorism, but no ocular anterior chamber, structural abnormalities have been reported until now. We evaluated three unrelated patients with severe Crouzon or Pfeiffer syndrome. Two of them had ocular findings consistent with Peters anomaly, and the third patient had opaque corneae, thickened irides and ciliary bodies, and shallow anterior chambers with occluded angles. Craniosynostosis with and without cloverleaf skull deformity, large anterior fontanelle, hydrocephalus, proptosis, depressed nasal bridge, choanal stenosis/ atresia, midface hypoplasia, and elbow contractures were also present. These patients had airway compromise, seizures, and two died by age 15 months. All three cases were found to have the same FGFR2 Ser351Cys (1231C to G) mutation predicted to form an aberrant disulfide bond(s) and affect ligand binding. Seven patients with isolated Peters anomaly, two patients with Peters plus syndrome, and three cases with typical Antley-Bixler syndrome were screened for this mutation, but none was found. These phenotype/genotype data demonstrate that FGFR2 is involved in the development of the anterior chamber of the eye and that the Ser351Cys mutation is associated with a severe phenotype and clinical course.
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keywords = airway
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