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A case of congenital lymphangiectasia of ventricular septum obstructing outflow tract is reported. It mimicks hypertrophic cardiomyopathy or a primary tumor of the heart. Surgical operation has been successful and the expected prognosis is good.
The Waldmann's disease or intestinal lymphangiectasia is a rare disease. We observed a case which occurred in a 14 years old boy. The disease was discovered in the screening of an intermittent diarrhea. The diagnosis was suspected on biological signs of protein-losing enteropathy and the presence of lymphangiectasias seen during endoscopy. The lymphography was conclusive showing the abnormal opacification of the mesenteric region. The other examinations were also important to assert the idiopathic origin of the disease.
Experiments performed on 71 Wistar rats confirm that preexisting interruption of lymph drainage by ligation of the ductus thoracicus can have a major influence on the development of pancreatitis. The effect of a ductus hepatopancreaticus blockade in experimental group A (32 animals) was greatly exacerbated by previous ligation of the ductus thoracicus (experimental group B; 34 animals). Edema of the interstitial pancreatic tissue led to lipolytic necrotizing pancreatitis with a slight hemorrhagic component, increasing ascites after the 12 th hour of the experiment, and numerous abdominal fat necroses after about 19 hours, but only relatively minor necroses of acinar parenchyma cells in the pancreas. Fat tissue necroses were only found in almost 20% of the animals in group A, and these probably resulted from manipulation of the duodenum during the implantation of a shunt to divert the bile, whereas they were found in all animals of group B after the 19th hour of the experiment, usually in large numbers. The sometimes considerable increases in serum amylase and particularly lipase activity were caused by obstruction of the efferent ducts, but did not increase appreciably after disturbance of the lymph drainage systems. On the contrary, under these circumstances, drainage of the salivary edema via the peritoneal mesothelium and into retroperitoneal fat tissue must be considered responsible for ascites and the initiation of fat cell necroses by lipase and other enzymes.
We describe a case of primary lymphatic dysplasia in a newborn, presenting with lymphedema, chylothorax, and chylous ascites. Malfunction of the lymphatic system, which is supposed to cause this rare disorder, could also be responsible for a number of cases of non-immunologic hydrops fetalis.
Massive ascites occurred as a complication of pelvic inflammatory disease. The patient underwent exploratory laparotomy with successful resolution of the ascites.
A case is reported of conception and successful completion of a pregnancy in a 31 year old woman maintained on home parenteral nutrition while suffering from a primary distension of the intestinal lymphatic vessels and intestinal loss of protein. A healthy, 3400 g female infant was born near term by cesarean section. This case confirms the few so far published experiences showing that conception and maintenance of the pregnancy under total parenteral nutrition needs no or just a slight adaptation of the regimen.
A patient with intestinal lymphangiectasia (IL) was studied for his immunological abnormalities. The patient had hypoproteinaemia with severe hypogammaglobulinaemia. The results of lymphocyte subpopulation studies revealed a decrease of CD4+ cells and a decrease of surface immunoglobulin (sIg)-positive B lymphocytes. T cell functions determined by the proliferative responses against Concanavalin A (Con A) or phytohaemagglutinin (PHA) and by delayed type cutaneous hypersensitivity (DTH) response to purified protein derivatives (PPD) and PHA were normal. No immunoglobulin (Ig)-secreting cells were induced when his peripheral blood lymphocytes (PBL) were cultured with pokeweed mitogen (PWM). His T cells showed suppressor function to the PWM-induced differentiation of normal B cells. This suppressor activity was sensitive to 3000 rads irradiation. His B cells also failed to differentiate into Ig-producing cells with the help of normal T cells and PWM. Thus, in this patient, the decrease of Ig-synthesis in vitro could be attributed to suppressor T cells, lack of T helper cells and an intrinsic B cell defect. Therefore, this patient appears to have immunological abnormalities which differ from previously reported IL patients.
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The term penicillamine dermatopathy refers to the characteristic hemorrhagic skin lesions found in persons receiving long-term penicillamine therapy for either Wilson's disease or cystinuria. These lesions are thought to develop as a result of faulty collagen and elastin synthesis. We describe a patient with Wilson's disease who developed extensive penicillamine dermatopathy. In addition, histologic, immunochemical, and ultrastructural studies revealed multiple lymphangiectases with blood vessel to lymphatic anastomosis within these lesions, a finding not previously reported. The possible relationship to defective collagen and elastin formation are considered.
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The experience in using radiolymphography in the diagnosis of central lymph dynamics disorders in thoracic duct pathology has been analysed. For the first time in Soviet literature we encounter two observations over the state of lymph dynamics along the thoracic duct by means of lymphoscintigraphy with colloid 113mIn in patients with lymphangiectasia of the thoracic duct and chylothorax. In importance of lymphoscintigraphy in determining the compensatory mechanisms of the lymph flow in primary pathology of the main lymph collector is emphasized.
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Numerous vascular dysplasiae belong to the group of genodysplasiae. Most arteriovenous dysplasiae (cirsoid, racemosum aneurysms,...) are the substratum of various regional "angiomatosis". Stenotic or ectatic arterial dysplasiae can be associated with genodysplasiae. In addition, lymphatic dysplasiae (congenital elephantiasis, etc...) also exist. Among benign tumours, angiomas are sometimes hardly distinguishable from dysplasiae. Some tumors with intermediate malignancy have an uncertain prognosis: chemodectoma, hemangiopericytoma. As for malignant vascular tumours, they fall into 3 varieties: angiosarcoma, Kaposi's sarcoma and sarcoma of vascular walls.
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