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PubMed1978

Transient intestinal lymphangiectasia.

Intestinal lymphangiectasia may be more protean in clinical manifestations and less rare than earlier suspected. A recent report points out that there are two types of the disorder, one congenital and the other acquired and transitory. A case is reported which fulfills the current clinical, laboratory, radiological and histological criteria for the diagnosis of the disease, and represents the first report in Scandinavia of transient intestinal lymphangiectasis with rapid and complete recovery within a few months after initiation of MCT diet.

Child↗
PubMed2000

[Familial Waldmann's disease].

We report the observation of a mother and her daughter who presented edema, hypoprotidemia and lymphopenia due to protein-losing enteropathy. Radiological, endoscopic and histological investigations revealed the diagnosis of primary intestinal lymphangiectasis or Waldmann's disease. Dietary treatment with middle chained triglycerides was effective. Familial cases are rarely described.

Adult↗
PubMedDate not supplied

[Intestinal lymphagiectasis. Use of immunoregulators].

The authors present a patient with intestinal lymphangiectasis discussing the pathophysiology, diagnosis and immunologic treatment instituted which was with mebendazole, sulfazalosopyridine and oral calcium. Immunoglobulins and T and B leukocytes showed increase together with clinical improvement in the diarrheal picture.

Benzimidazoles↗
PubMed1981

Resolution of longstanding protein-losing enteropathy in a patient with intestinal lymphangiectasia after treatment for malignant lymphoma.

In 1956 we evaluated a patient who had a debilitating disease of a 2 yr duration, characterized by recurrent vomiting, diarrhea, cachexia, massive edema, hypoproteinemia, and dilated intestinal lymphatics. During our initial evaluation of this patient, we observed that 42% of her circulating protein pool was lost into her gastrointestinal tract daily, whereas normal gastrointesinal loss of protein does not exceed 1.6%. Her disease appeared to represent a classic example of intestinal lymphangiectasia. She was treated symptomatically for 13 yr with essentially no change. In 1969 the patient developed a stage IV diffuse, undifferentiated (non-Burkitt's) malignant lymphoma. Using immunoperoxidase staining, the neoplastic cells were found to contain cytoplasmic IgMKappa, suggesting that the lymphoma had a monoclonal B-cell origin. She was successfully treated with cyclophosphamide, vincristine, and prednisone. Shortly after the initiation of this systemic combination chemotherapy, her serum protein concentration returned to normal, her edema resolved, and she was cured of gastrointestinal symptoms. Moreover, repeat studies revealed that her protein loss had fallen to only 2%. The simultaneous cure of both the intestinal lymphangiectasia and lymphoma with combination chemotherapy suggests new relationships between these conditions as well as new possibilities for the treatment of acquired forms of intestinal lymphangiectasis associated with overwhelming gastrointestinal protein loss.

Adult↗
PubMed2000

[Waldmann disease and pregnancy].

Waldman disease, also called intestinal lymphangiectasis, is a protein-loosing enteropathy caused by anomalous intestinal lymphatic canals. The condition is transmitted by autosomal dominant inheritance. Diet therapy including medium chain triglycerides improves the disease course. The condition occurs in early childhood. We report a case observed in a pregnant woman with intestinal lymphangiectasis. She delivered a full-term infant and experienced no major complications.

Adult↗
PubMed1976

[Retroperitoneal lymphangiodysplasia with exudative enteropathy. A case of cystic dilated cisterna chyli and splenic cysts].

In a 16 years old girl we found the syndrome of exsudative enteropathy with intestinal lymphangiectasy which differs from previous reported cases by several malformations of the lymphatic system. Besides changes in the intestinal lymphatics there was a retroperitoneal lymphangiodysplasia, a reduction and hypoplasia of the lymph vessels in the legs and cysts in the spleen. By substitution of albumin followed by a diet with middle chained triglycerids (MCT), serum protein was stablised but a relapse could not be prevented. Surgical treatment was not possible because the changes were too extensive. The value of diagnostic measures for differential diagnosis of the intestinal lymphangiectasy is presented.

Adolescent↗
PubMed2000

Computed tomography after lymphangiography in the diagnosis of intestinal lymphangiectasia with protein-losing enteropathy in Noonan's syndrome.

Noonan's syndrome is a rare congenital disorder that may be associated with abnormalities in the lymphatic drainage. In this case of a 21-year-old man CT after bipedal lymphangiography confirmed the diagnosis of intestinal lymphangiectasy causing protein-losing enteropathy in Noonan's syndrome by showing contrast-enhanced abnormal lymphatic vessels in the mesentery and the intestinal wall. Because of the benefit of diet in case of intestinal involvement, we recommend a thorough documentation of the lymphatic drainage with lymphangiography followed by CT, if clinical signs of lymphatic dysplasia, such as pleural effusions, lymphedema, or hypoproteinemia are present.

Adult↗
PubMedDate not supplied

[Diagnostic value of jejunal biopsy].

Trying to find out the clinical elements that define the precise indication ofr a jejunal biopsy, 28 children with several pathological conditions were studied. They were 12 cases of kwashiorkor, 11 cases with chronic diarrhea, two cases of chronic pancreatitis, 2 cases with ferropenic anemia resistant to the oral treatment with iron and one case of chilous ascitis. It is concluded that only in those cases in which the biopsy is the precise medium for diagnosis is where it would be indicated, such as intestinal lymphangiectasis or in those cases with signs or evidence of malabsorption without diarrhea. The chronic diarrhea per se does not seem to be a formal indication for biopsy.

Adolescent↗
PubMed1986

Nonimmune hydrops fetalis in Noonan's syndrome.

Nonimmune hydrops fetalis has been reported to be associated with congenital malformations. We describe two newborns with Noonan's syndrome who presented with nonimmune hydrops fetalis that was most likely secondary to a generalized lymphatic vessel dysplasia. Other manifestations of lymphatic abnormalities in Noonan's syndrome, such as pedal edema and pulmonary and intestinal lymphangiectasis, have been observed in children. Nonimmune hydrops represents one end of the spectrum of abnormalities seen in this syndrome.

Edema↗
PubMed2001

Isolated fetal ascites caused by primary lymphangiectasia: a case report.

We present a case of isolated fetal ascites diagnosed by ultrasonography at 29 weeks' gestation. Cordocentesis revealed a normal karyotype and negative viral titers. Postnatally, the diagnosis of primary lymphangiectasia was made by intestinal biopsy. To our knowledge this is the first description of prenatal manifestation of primary lymphangiectasia.

Adult↗
PubMed1983

Lymphoscintigraphic demonstration of intestinal lymphangiectasia.

The authors report a case of intestinal lymphangiectasia that was clearly demonstrated during abdominal lymphoscintigraphy using Tc-99m Sb colloid. Although lymphoscintigraphy has been used in many different clinical situations, this is, to our knowledge, the first scintigraphic description of this disease. Clinical and pathologic presentations of intestinal lymphangiectasia are briefly reviewed. Some possible approaches to the confirmation of the presence of a protein-losing gastroenteropathy are mentioned.

Adult↗