查看更多>>摘要：Lymphangioma is a rare benign condition characterized by proliferation of lymphatic spaces.It is usually found in the head and neck of affected children.Lymphangioma of the small-bowel mesentery is rare,having been reported for less than 1％ of all lymphangiomas.Importantly,it can cause fatal complications such as volvulus or involvement of the main branch of the mesenteric arteries,requiring emergency surgery.Moreover,the gross and histopathologic findings may resemble benign multicystic mesothelioma and lymphangiomyoma.Immunohistochemical study for factor Ⅷ-related antigen,D2-40,calretinin and human melanoma black-45 (HMB-45) are essential for diagnosis.Factor Ⅷ-related antigen and D2-40 are positive in lymphangioma but negative in benign multicystic mesothelioma.HMB-45 shows positive study in the smooth-muscle cells around the lymphatic spaces of the lymphangiomyoma.We report a case of smallbowel volvulus induced by mesenteric lymphangioma in a 2-year-and-9-mo-old boy who presented with rapid abdominal distension and vomiting.The abdominal computed tomography scan showed a multiseptated mass at the right lower quadrant with a whirllike small-bowel dilatation,suggestive of a mesenteric cyst with midgut volvulus.The intraoperative findings revealed a huge,lobulated,yellowish pink,cystic mass measuring 20 cm × 20 cm × 10 cm,that was originated from the small bowel mesentery with small-bowel volvulus and small-bowel dilatation.Cut surface of the mass revealed multicystic spaces containing a milky white fluid.The patient underwent tumor removal with small-bowel resection and end-to-end anastomosis.Microscopic examination revealed that the cystic walls were lined with flat endothelial cells and comprised of smooth muscle in the walls.The fiat endothelial cells were positive for factor Ⅷ-related antigen and D2-40 but negative for calretinin.HMB-45 showed negative study in the smooth-muscle cells around the lymphatic spaces.Thus,the diagnosis was lymphangioma of the small bowel mesentery with associated small bowel volvulus.
查看更多>>摘要：A number of disorders have been described to cause protein losing enteropathy (PLE) in children.Primary intestinal lymphangiectasia (PIL) is one mechanism leading to PLE.Few syndromes are associated with PIL; Hennekam syndrome (HS) is one of them.The principal treatment for PIL is a high protein,low fat diet with medium chain triglycerides supplementation.Supportive therapy includes albumin infusion.Few publications have supported the use of octreotide to diminish protein loss and minimize hypoalbuminemia seen in PIL.There are no publications on the treatment of PIL with octreotide in patients with HS.We report two children with HS and PLE in which we used octreotide to decrease intestinal protein loss.In one patient,octreotide increased serum albumin to an acceptable level without further need for albumin infusions.The other patient responded more dramatically with near normal serum albumin levels and cessation of albumin infusions.In achieving a good response to octreotide in both patients,we add to the publications supporting the use of octreotide in PIL and suggest that octreotide should be tried in patients with PIL secondary to HS.To the best of our knowledge,this is the first case report on the use of octreotide in HSassociated PIL.
查看更多>>摘要：Colonic intussusception and gastrointestinal duplication are diseases that arise in young children.The clinical presentation of adult cases of intussusception and enteric duplication is non-specific and thus poses a diagnostic challenge.A computed tomography (CT) scan is recommended in adult cases as the most sensitive diagnostic tool and the pathognomonic finding of outer intussuscepiens and central intussusceptum is diagnostic.A septum of a duplicated colon in a non-intussuscepted segment has been rarely reported in the literature.With advancements in radiological imaging technology and the increased availability of CT scanners,the capacity for a correct pre-operative diagnosis has been significantly enhanced.Our current case report illustrates the importance of considering an uncommon etiology for enteric intussusception and duplication as a differential diagnosis of acute abdomen in an adult patient.Our analyses of this patient also highlight the successful use of CT scanning to make this diagnosis.
查看更多>>摘要：An isolated parenchymal gastric metastasis from ovarian carcinoma without any other sites of recurrence is extremely rare.Only two cases have been reported,both of which were symptomatic.We herein report such a case without any symptoms.A 61-year-old woman presented with a high cancer antigen-125 level without any other clinical manifestation.A subsequent 18F-fluorodeoxyglucose (18F-FDG) positron emission tomography/computed tomography scan revealed a submucosal mass with hypermetabolism of 18F-FDG (standardized uptake value:5.36) in the gastric antrum.The final pathology after gastric antrectomy showed a metastatic gastric tumor from a primary ovarian carcinoma.We also performed an extensive literature review about gastric metastasis from ovarian carcinoma published until recently,and this is the first case of an isolated parenchymal gastric metastasis from ovarian carcinoma without any symptoms.
查看更多>>摘要：The expression of phosphatase and tensin homolog (PTEN),a tumor suppressor gene,is frequently downregulated in gastric carcinomas due to mutation,loss of heterozygosity,and promoter hypermethylation.However,it is unknown if additional mechanisms may account for the down-regulation of PTEN expression.While neuronal precursor cell-expressed developmentally down-regulated 4-1 (NEDD4-1) is believed to be a potential dual regulator of PTEN,there are conflicting reports regarding their interaction.To gain further insight into the role of NEDD4-1 and its association with PTEN in gastric carcinoma development,we measured the protein expression of NEDD4-1 and PTEN in gastric mucosae with various pathological lesions and found that NEDD4-1 increased from normal gastric mucosa to intestinal metaplasia and decreased from dysplasia to gastric carcinoma.These changes did not correlate with PTEN expression changes during gastric carcinogenesis.Moreover,we found similar results in protein levels in the primary tumors and adjacent non-tumorous tissues.These results differ from a previous report showing that expression of NEDD4-1 is up-regulated in gastric carcinomas,and show a more complex pattern of NEDD4-1 gene expression during gastric carcinogenesis.
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