Cases reported "Disease Models, Animal"

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1/8. Single-vessel arteriovenous revascularization of the amputated ear.

    Successful single-vessel arteriovenous replantation of a completely amputated human ear is described. This result was followed by an experimental study using a rabbit model, to confirm that an amputated ear could survive replantation with only a single arteriovenous anastomosis. Fifteen animals were placed in one of two experimental groups: Group 1-arteriovenous replantation, no treatment (n = 6); and Group 2-arteriovenous replantation with medicinal leeching (n = 9). All ears demonstrated initial reperfusion of the replantation immediately following microanastomosis. Laser Doppler flow readings in the non-leeched replanted ears fell to zero by 8 hr and, by 12 hr, the non-leeched ears demonstrated signs of necrosis. All the leeched, replanted ears demonstrated perfusion and complete viability at the time of sacrifice. The case report, combined with the results from the experimental study, confirm that single-vessel arteriovenous replantation of an amputated ear is feasible.
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2/8. Can early postnatal closed head injury induce cortical dysplasia.

    PURPOSE: Increased availability of surgically resected epileptogenic tissues reveals often unsuspected cortical dysplasia (CD). There is some controversy about the ontogenic stages in which these occur. Although most take place during neuroblast proliferation and migration, there is some evidence for some CD occurring during postmigrational intrinsic cortical organization. It has been shown that various kinds of focal cortical manipulations in rats, if performed within 3-4 postnatal days, lead to the genesis of various cortical malformations including a four-layered microgyrus or an unlayered CD. It is not known whether such events also might occur in the human brain. methods: Two children sustained minor head trauma within 4 postnatal days and later developed intractable epilepsy, which was relieved by surgery. Neuropathologic analysis of the resected tissues revealed an unsuspected microdysplastic cortex immediately adjacent to a focal, modest meningeal fibrosis, presumably secondary to the old closed head trauma. RESULTS: The main histologic features were a disorganized, unlayered cortex; abnormal clusters of neurons, often with complex, randomly oriented proximal dendritic patterns with absent apical orientation; the presence of a number of heterotopic small and large neurons in the white matter; absence of inflammatory infiltrates, of hemosiderine, of reactive gliosis, or of an excessive number of blood vessels. The morphologic features in these surgical specimens suggest that these focal malformations occur because of a regional disorder of postmigrational intrinsic cortical remodeling. CONCLUSIONS: The clinical histories and the pathologic findings lend some support to the hypothesis that minor morbid events occuring in the immediate postnatal period may lead to microdysplasia in the human similar to those induced in rat pups. The animal model could be helpful to clarify the genesis of some cases of CD and of the epileptogenicity often manifesting later in life.
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3/8. Abnormality of vascular elastic fibers in the macular mouse and a patient with Menkes' disease: ultrastructural and immunohistochemical study.

    The macular mouse is a mutant mouse with the same gene abnormality as that of Menkes' disease, and it exhibits symptoms and abnormalities similar to those of Menkes' disease. In an electron microscopic study, we examined morphological changes in the internal elastic lamina (IEL) of the elastic arteries (EA) and the muscular arteries (MA) in a patient with Menkes' disease and in the macular mouse, an animal model of this disease. The IEL of the EA was significantly thinner in the macular mouse than that in controls, but the IEL of the MA in the macular mouse was significantly thicker than that of the controls. These contrary results for the thickness of the IEL in the MA and the EA in this animal model of Menkes' disease may reflect differences in the anatomical and pathophysiological properties of the two types of vessels.
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4/8. octreotide in intestinal lymphangiectasia: lack of a clinical response and failure to alter lymphatic function in a guinea pig model.

    Intestinal lymphangiectasia, which can be classified as primary or secondary, is an unusual cause of protein-losing enteropathy. The main clinical features include edema, fat malabsorption, lymphopenia and hypoalbuminemia. Clinical management generally includes a low-fat diet and supplementation with medium chain triglycerides. A small number of recent reports advocate the use of octreotide in intestinal lymphangiectasia. It is unclear why octreotide was used in these studies; although octreotide can alter splanchnic blood flow and intestinal motility, its actions on lymphatic function has never been investigated. A case of a patient with intestinal lymphangiectasia who required a shunt procedure after failing medium chain triglycerides and octreotide therapy is presented. During the management of this case, all existing literature on intestinal lymphangiectasia and all the known actions of octreotide were reviewed. Because some of the case reports suggested that octreotide may improve the clinical course of intestinal lymphangiectasia by altering lymphatic function, a series of experiments were undertaken to assess this. In an established guinea pig model, the role of octreotide in lymphatic function was examined. In this model system, the mesenteric lymphatic vessels responded to 5-hydroxytryptamine with a decrease in constriction frequency, while histamine administration markedly increased lymphatic constriction frequency. octreotide failed to produce any change in lymphatic function when a wide range of concentrations were applied to the mesenteric lymphatic vessel preparation. In conclusion, in this case, octreotide failed to induce a clinical response and laboratory studies showed that octreotide did not alter lymphatic function. Thus, the mechanisms by which octreotide induced clinical responses in the cases reported elsewhere in the literature remain unclear, but the present study suggests that it does not appear to act via increasing lymphatic pumping.
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5/8. Renal failure following radiologic procedures.

    Radiologic procedures that employ intravascular contrast material with or without angiography may lead to renal failure. In procedures that use intravenous contrast alone, the mechanism of renal injury is not precisely known, but direct toxicity to renal tubular cells is likely to be a major factor. Ionic and nonionic contrast agents are both capable of causing this adverse reaction. Renal failure occurring during angiography may also be secondary to the effects of radiocontrast, but the additional possibility that micro cholesterol emboli have been dislodged from atheroma located on the intima of large vessels must be considered. The acute or subacute development of renal failure in the presence of skin changes (livido reticularis), hypertension, multiple organ failure or dysfunction, and a fatal outcome favors the later diagnosis.
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6/8. Hazards of calcium gluconate therapy in the newborn infant: intra-arterial injection producing intestinal necrosis in rabbit ileum.

    Five infants received 10% calcium gluconate via umbilical artery catheters, which resulted in intestinal bleeding and lesions of the buttock, anus, groin, and thigh. The effects of intra-arterial calcium gluconate in two animal models were investigated. Injection of calcium into the aorta in the region of the posterior mesenteric artery resulted in immediate hyperperfusion of the descending colon; this may be an early hemodynamic response to injury in the area of colon supplied by this vessel. injections into the arterial arcade of the rabbit ileum resulted in intestinal necrosis and villous atrophy. The use of umbilical artery catheters for administration of calcium gluconate is potentially hazardous.
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7/8. Lymphatic vessel-to-isolated-vein anastomosis for secondary lymphedema in a canine model.

    To design a more rational and effective surgical method of performing lymphatic-venous anastomosis to treat secondary lymphedema of the lower extremities, the following experiments were conducted on three groups of dogs: group A underwent an end-to-side lymphatic node-to-vein anastomosis at the inferior vena cava; group B underwent a "burying" lymphatic vessel-to-vein anastomosis at the femoral vein; and group C underwent a burying lymphatic vessel-to-isolated-vein anastomosis at the femoral vein. In group C, the femoral venous segment was isolated by distal ligation and proximal valvuloplasty and the patency of the anastomosis was investigated by infusing yellow Microfils through the distal lymphatic vessel. The patency of the anastomosis was nil in group A by 10 days after the anastomosis, 40% in group B by 180 days; and 71.4% in group C by 180 days, respectively. Thus, we clinically applied the technique of lymphatic vessel-to-isolated-saphenous-vein anastomosis in a patient with secondary lymphedema of the bilateral lower extremities. A satisfactory reduction in the size of the limbs was achieved and there has been no further recurrence of cellulitis in the 42 months since her surgery. This study shows that lymphatic vessel-to-vein anastomosis is an effective technique for the surgical management of secondary lymphedema, so long as the anastomosis is completely protected from any contact with blood.
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8/8. Use of intravascular stents in the treatment of internal carotid and extracranial vertebral artery pseudoaneurysms.

    The management of extracranial carotid or vertebral artery pseudoaneurysms is controversial. Although some of these lesions resolve spontaneously, many clinicians opt to treat them with trapping procedures that result in vessel sacrifice. We describe two cases in which an intravascular stent was used to obliterate an aneurysm of the extracranial vertebral artery and the internal carotid artery, respectively, while maintaining the patency of the parent vessel. The technique, which has been successful in experimental animal models, shows promise for application in humans.
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