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PubMed1977

[Electron microscopic study of the small intestine in intestinal lymphangiectasia and constrictive pericarditis (author's transl)].

Electron microscopic study of the small intestine in cases of intestinal lymphangiectasia and in cases of constrictive pericarditis was performed. This study revealed that numerous chylomicron-like particles are present in the lymphatic lumina, in the extracellular spaces of the lamina propria, and within the interepithelial spaces between the absorptive cells. Presence of chylomicron-like particles in the intestinal lumen suggested possibility of passage of these substances into the intestinal tract through the interepithelial spaces. Similar findings to those as seen in intestinal lymphangiectasia were observed in constrictive pericarditis. A few pseudopode-like cytoplasmic projections of the undifferentiated crypt cells were noted. Other mechanisms of enteric protein loss are postulated.

Chylomicrons↗
PubMed1993

Fat absorption in patients with functional intestinal lymphangiectasia and lymphangiectic cysts.

Nine patients with endoscopically identified dilated lacteals of the duodenum were studied for evidence of pathologic intestinal lymphangiectasia. Three of the nine patients also had lymphangietic cysts in association with dilated lacteals. Duodenal biopsies, laboratory data, and imaging studies were performed in each patient. In addition, a 14C triolein fat absorption study was performed to assess subclinical malabsorption. Biopsies revealed dilated lymphatic channels in all patients, but laboratory studies failed to suggest intestinal losses of protein or fat, and radiographic abdominal imaging failed to define any of the causes of secondary lymphangiectasia. Eight of the nine patients had adequate fat absorption as measured by the 14C triolein breath test. Our data suggest that patients with incidentally discovered dilated lacteals and no clinical evidence of malabsorption may have a functional intestinal lymphangiectasia. Follow-up endoscopy probably is not warranted in this population.

Biopsy↗
PubMed1996

Intestinal leakage of technetium-99m-MDP in primary intestinal lymphangiectasia.

We present a case in which a patient with primary intestinal lymphangiectasia demonstrated abnormal intestinal accumulation of tracer during 99mTc-methylene diphosphonate (MDP) skeletal scintigraphy. Early intestinal leakage with gradual colonic migration and concentration was confirmed by repeat bone scan with serial acquisitions. The mechanism for the intestinal localization of 99mTc-MDP seen in this patient is not clear. Thus, intestinal lymphangiectasia can be a cause for extra-osseous localization of bone scan agents in the intestine.

Adult↗
PubMed1979

[The diagnostic value and risks of small bowel biopsy: an assessment in 80 patients (author's transl)].

Small bowel biopsy is a routine procedure used mainly in the evaluation of malabsorption. The Crosby-Kugler capsule, which permits the performance of just one biopsy per session and hydraulic biopsy tubes, permitting multiple biopsies from any level of the small bowel, have been in use now with several modifications for about 20 years. The diagnostic range and risks of these tools are described briefly in this paper and discussed in the light of our own experience in 80 patients. In accordance with the experience of others, we found that small bowel biopsy was accompanied by only a few minor complications and we consider it a valuable procedure in clinical diagnosis and experimental research.

Biopsy↗
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↗
PubMed1996

An unusual presentation of intestinal lymphangiectasia.

Intestinal lymphangiectasia is a rare disorder, usually presenting with protein-losing enteropathy leading to oedema and ascites. We describe a case that presented as an acute surgical emergency with pain and a mass in the right iliac fossa, which was successfully treated by resection of the involved segment of ileum.

Abdomen, Acute↗
PubMed2006

Videocapsule endoscopy is useful for the diagnosis of intestinal lymphangiectasia.

We study two authentic cases of protein-losing enteropathy, the diagnosis of which was facilitated using Given M2A videocapsule endoscopy. The first case corresponded to a primary intestinal lymphangiectasia confirmed by jejunum biopsies and the second one to a protein-losing enteropathy with lymphatic abnormalities secondary to a chronic constrictive pericarditis. In the first case, the mucosa of jejunum presented with a diffuse oedematous aspect, whitish villi, white curved lines probably related to submucosal dilated lymphatics and lacteal juice. In the second case, capsule endoscopy showed oedematous aspect of jejunum mucosa associated with white curved lines similar to those observed in the first case. Videocapsule endoscopy is useful in cases of protein-losing enteropathy to identify presence of intestinal lymphangiectasia and to specify their localisation after ruling out other disorders liable to induce protein-losing gastrointestinal syndrome.

Adult↗
PubMed1979

Intestinal lymphangiectasia and colonic polyps: surgical intervention.

A 36-mo-old boy with Milroy's Disease, intestinal lymphangiectasia, and an exudative enteropathy (EE), was shown to have four colonic polyps. A large adenomatous polyp was excised from the transverse colon in an effort to control his EE and hypoalbuminemia (1.95 g/dl). His clinical status then stabilized until age 50 mo when there was a marked exacerbation of his EE. Medical management resulted in a temporary stabilization of his condition. A partial resection (40 cm) of the visually worse affected jejunum was performed. There was no improvement in the EE as measured by 51Cr-tagged albumin study; however, his clinical response was dramatic. In the 10 mo since surgery, he has been well and has shown catchup in linear growth.

Child, Preschool↗
PubMed1998

Ultrasonographic features of long-standing primary intestinal lymphangiectasia.

This article describes a case of primary intestinal lymphangiectasia associated with protein-losing enteropathy and lower right leg lympoedema in a 20-year-old woman. Echographic findings showed dilation of the intestinal loops, regular and diffuse thickening of the walls, plical hypertrophy and impressive mesenteric oedema. Although diagnosis of the disease is substantially histological, the ultrasonographic characteristics of the lesions are fairly indicative and of certain diagnosis usefulness in patients with protein-losing enteropathy.

Adult↗
PubMed1975

Limitations of the usefulness of the d-xylose absorption test.

The results of the conventional 5-hour d-xylose absorption test were surveyed in 38 subjects with disease of the jejunal mucosa, giardiasis or bacterial overgrowth, or no small-bowel disorder. The test was in error in 20-40% of cases, depending on the disease category, and the error for the entire group was 30%. We conclude that the test yields little guidance for diagnosis or therapy of clinical problems and is superfluous when a jejunal biopsy can be obtained.

Administration, Oral↗
PubMed2001

Protracted diarrhea: results of the five-year survey in a tertiary hospital in Korea.

The syndrome of protracted diarrhea (PD) includes several diseases with diverse etiologies. This study was conducted to characterize the spectrum of causes, clinical manifestations, and the outcomes of PD. A retrospective analysis of the clinical and pathological findings was performed on 25 patients with diarrhea starting within the first 2 yr of life and a requirement of parenteral nutrition (PN). According to the intestinal histopathology, patients were classified into four groups: immune enteropathy (12 cases), lymphangiectasia (6 cases), epithelial dysplasia (5 cases), and unclassified (2 cases). All patients with epithelial dysplasia had earlier onset of diarrhea and longer duration of PN than those in the other groups. Three patients (12%) had an evidence of a familial condition. Five patients (three with microvillous inclusion disease and two with immune enteropathy) died. Sixteen patients recovered, and three (two with primary lymphangiectasia and one with microvillous inclusion disease) still had diarrhea. One patient underwent intestinal transplantation for tufting enteropathy. In conclusion, infants with PD should be referred to specialized centers where advanced diagnostic and therapeutic facilities are available, because histological analysis is critical for the diagnosis of PD, and PN or intestinal transplantation is the only therapeutic option in a subset of cases.

Academic Medical Centers↗
PubMed1975

[Tumoral extension by inversion of the lymphatic flow in colonic cancer].

In a patient with an adenocarcinoma of the ascending colon, the jejunal central villous lymphatics were dilated and contained tumour cells. This case, apparently the only one of its type, demonstrates the proof of a phenomenon sometimes described in connection with malignant diseases, but rarely proven; that of the dissemination of tumour cells by reversal of lymphatic flow.

Adenocarcinoma↗