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PubMed1987

Intestinal lymphangiectasia secondary to radiotherapy and chemotherapy.

We report a case of intestinal lymphangiectasia secondary to radiotherapy and chemotherapy. The patient also had small bowel bacterial overgrowth and pancreatic insufficiency. Lymphatic ectasia as a histological feature has been described previously in association with postradiotherapy malabsorption, but radiation-induced lymphangiectasia producing clinical manifestations has hitherto not been reported. Replacement of dietary long-chain fats with medium-chain triglycerides, pancreatic enzyme supplements, and a short course of oxytetracycline, resulted in dramatic clinical improvement. The possibility of intestinal lymphangiectasia should be borne in mind in patients with postradiotherapy malabsorption. A low serum albumin and lymphocyte count should draw attention to this possibility.

Adult↗
PubMed1981

Endoscopic and histopathological study on primary and secondary intestinal lymphangiectasia.

Jejunal endoscopy and histopathological study of biopsied specimens were performed to clarify states of jejunal mucosa and the mechanism of enteric protein loss in six patients with protein-losing enteropathy, including four patients with intestinal lymphangiectasia, one patient with constrictive pericarditis associated with dilated lymphatics of the intestine, and one patient with Budd-Chiari syndrome. Three cardinal endoscopic findings, scattered white spots, white villi, and chyle-like substances covering the mucosa, were demonstrated in protein-losing enteropathy. Scattered white spots indicated markedly dilated lymphatics in the stroma of the villi. White villi seemed to be due to fats including chylomicrons or fat droplets in the absorptive cells, interepithelial spaces, and/or stroma, even though the biopsies were obtained in the fasting state. Therefore, white villi suggest impaired transport of fats from intestinal epithelial cells to intestinal lymphatics. These three cardinal findings are thought to be characteristic for protein-losing enteropathy secondary to lymphatic disorders.

Adolescent↗
PubMed1992

Intestinal lymphangiectasia: evaluation by CT and scintigraphy.

Intestinal lymphangiectasia caused severe diarrhea and generalized edema in a 40-year-old man. The diagnosis was established by clinical, laboratory, and duodenal biopsy findings. The abnormalities detected on computed tomography (CT) and scintigraphy using 99mTc human serum albumin are herein described and pertinent literature is briefly reviewed.

Adult↗
PubMed1995

A radiologic update on medical diseases of the newborn chest.

This paper reviews the common spectrum of medical diseases of the neonatal chest. Emphasis is on radiographic changes that have been produced by the introduction of new therapeutic maneuvers, particularly the use of artificial surfactant in treating hyaline membrane disease and the survival of profoundly premature newborns (less than 650 g). A discussion of meconium aspiration syndrome, neonatal pneumonia, transient tachypnea of the newborn, congenital lymphangiectasia, and congenital heart disease is also included. The effects on the neonatal chest radiograph of extracorporeal membrane oxygenation and high-frequency ventilation are also mentioned.

Heart Defects, Congenital↗
PubMed1994

Pathogenesis of varices in schistosomal portal hypertension.

Patients with schistosomiasis and portal hypertension have significantly lower levels of hydroxyproline in their saphenous veins and anterior rectus sheaths than do individuals without schistosomal hepatic fibrosis. The stomach of patients with schistosomal portal hypertension demonstrates an increased number of lymphatics by lymphangiography. The disrupted lymph node architecture in these patients could be partially responsible for dilation, tortuosity, and retrograde lymph flow in the gastric lymphatics. These histological and lymphangiographic findings could be attributed to the effect of venous and patients with decompensated schistosomal portal hypertension revealed edema of the entire esophageal wall with lymphatic dilation and tortuosity. Based upon these data, we suggest that the varices that develop in patients with schistosomal portal hypertension occur as a consequence of an increased portal venous pressure together with acquired lymphangiectasia as well as an intrinsic weakness of the walls of the portosystemic venous channels.

Collagen↗
PubMed1986

Fetal lymphangiectasia--another cause for a positive amniotic fluid acetylcholinesterase test.

A case of a fetal lymphangiectasia on the ventral aspect of the neck was detected prenatally by ultrasound examination. Polyhydramnios was present. AFP values in amniotic fluid and maternal serum were in the upper normal range. An acetylcholinesterase band was found on gel electrophoresis. After delivery the prenatal diagnosis of congenital isolated lymphangiectasia was confirmed. The case illustrates a further example of a positive acetylcholinesterase test not associated with a neural tube defect.

Acetylcholinesterase↗
PubMed1976

Lymphangiectasia of the small intestine: description and pathophysiology of the roentgenographic signs.

Waldmann in 1961 redefined primary protein-losing gastroenteropathy and renamed the condition "lymphangiectasia". This abnormality, usually seen in infancy, is characterized by enlargement of folds and signs of hypersecretion in the small bowel. Enlargement of folds occurs secondary to edema of the valvulvae conniventes and lymphatic dilatation. Hypersecretion may be the result of rupture of dilated lymphatics or transudation of protein across an intact capillary epithelium. The clinical, roentgenographic, and pathologic findings are described. The pathophysiology of this condition is discussed.

Diagnosis, Differential↗
PubMed1987

Two cases of Winchester syndrome: with increased urinary oligosaccharide excretion.

We present our findings in two unrelated patients with the characteristic clinical and radiological features of the Winchester syndrome. The histological findings in gum and skin biopsies taken from one of the subjects, indicated excessive collagen turnover (active phagocytosis, an active endoplasmic reticulum, and an abundance of fibrillogranular material of probable collagen origin). An abnormal oligosaccharide was detected in urine from both patients which was identified as a trisaccharide containing one fucose and two galactose residues. The finding of this oligosaccharide may prove a useful marker in other cases of this rare syndrome and may help elucidate the underlying biochemical defect.

Child↗
PubMed1980

Pathological and pathophysiological study on intestinal lymphatic system in fat absorption.

Pathological and pathophysiological study on the intestinal lymphatics was carried out clinically and experimentally. Jejunal biopsy study revealed marked dilatation of intestinal lymphatics in liver cirrhosis with ascites and Behçet's disease as well as protein-losing enteropathy. Lymphangiographic study showed also abnormal findings in above-mentioned diseases. In cases of McKee dogs in which mechanism of blockade of lymphatic flow was already clarified, McKee dogs showed marked malabsorptive of fat. When lymphatic flow was blocked mechanically at the thoracic duct, fat accumulation was seen in the absorptive cells as well as in dilated intestinal lymphatics. In administration of colchicine to rats, fat accumulation was seen in the matrix of absorptive cells as well as in the endoplasmic reticulums and Golgi apparatus. However, amorphous substances instead of chylomicrons were seen in dilated intestinal lymphatics. These studies suggested that a tract of fat transportation from intestinal cells to the thoracic duct should be considered as a functional unit in fat absorption.

Animals↗
PubMed1976

Comparative, immunological studies on lymphangiectasia of the small intestine revealed in protein losing gastroenteropathy and Behçet's disease.

Lymphangiectasia of the small intestine was demonstrated in 9 to 18 cases with protein losing gastroenteropathy and in 10 of 26 cases with Behçhet's disease. Protein losing gastroenteropathy was compared to Behçet's disease in view of immunological aspects. Immunoglobulin containing cells in the jejunal mucosa of protein losing gastroenteropathy were decreased, whereas Behçet's disease had normal or increased distribution. This suggested that suggested that immunoglobulin may be lost into the intestinal lumen or that production of those cells may be inhibited in protein losing gastroenteropathy. Decreased DNCB skin reaction and impaired blastoid transformation of peripheral lymphocytes in both of two diseases suggested that there should be immunological defect in those diseases. However, in Behçet's disease, investigation of the thymus disclosed hyperplasia and the presence of lymphoid follicle which does not appear in normal state. In conclusion, protein losing gastroenteropathy is in immunological deficiency state due to the congenital or acquired disorders of lymphatics which implies abnormal protein loss, decreased immunoglobulin containing cells in the jejunal mucosa and decreased sensitivity. On the contrary, in Behçet's disease hyperimmune state is present based on thymic hyperplasia.

Adult↗
PubMed1993

Constrictive pericarditis simulating intestinal lymphangiectasia in a patient with the Noonan syndrome.

A case of constrictive pericarditis with marantic endocarditis in a patient with the Noonan syndrome is reported. Congenital heart defects are often diagnosed in the Noonan syndrome and are undoubtedly the most problematic of its pathologies. Our patient had surgery for pulmonic stenosis at age 10 and 16 years. Over a period of 1-2 years prior to death at age 23 years, he developed elevated jugular venous pressure, hypoproteinaemia, pedal oedema and pleural effusions. The hypoproteinaemia and resulting signs were initially attributed to intestinal lymphangiectasia. The latter, unlike constrictive pericarditis, has been reported in the Noonan syndrome. Post-mortem examination revealed constrictive pericarditis with a marantic endocarditis. There was no evidence of intestinal lymphangiectasia.

Adult↗
PubMedDate not supplied

Common pulmonary vein atresia: a diagnostic and therapeutic challenge.

Following Doppler echocardiographic evaluation, a 16 hour-old infant underwent successful surgical repair of common pulmonary vein atresia. Investigations for prolonged postoperative ventilatory assistance, including cardiac catheterization and computerized tomography, led to a clinical diagnosis of associated pulmonary lymphangiectasia. Although this has caused continuing tachypnoea, at 16 months of age, he remains otherwise well but with an uncertain prognosis.

Cardiac Catheterization↗