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PubMed2001

Different patterns of lymphoscintigraphic findings in patients with intestinal lymphangiectasia.

Technetium-99m antimony sulfide colloid lymphoscintigraphy conveniently demonstrates intestinal leakage of lymph in patients with intestinal lymphangiectasia. However, we found no intestinal radioactivity in some patients. We evaluated lymphoscintigraphic findings and compared them with clinical data. Technetium-99m antimony colloid lymphoscintigraphy was performed in 12 patients (age, 8.9+/-6.4 years; male:female=8:4) with histologically proven intestinal lymphangiectasia. After subcutaneous injection of 103.6 MBq of technetium-99m antimony colloid into the webs of both feet, sequential abdominal images were obtained up to 24 h post-injection. Four patients underwent technetium-99m methylene diphosphonate bone scintigraphy. Patients were divided into two groups according to the presence or absence of intestinal radioactivity. Five showed intestinal activity (Group 1), but seven did not (Group 2). No Group 1 patient had a history of ascites, while all Group 2 patients had ascites as the initial manifestation. Serum total protein and albumin levels were significantly lower in Group 1 patients than in Group 2 patients. In three Group 1 patients, technetium-99m methylene diphosphonate bone scintigraphy revealed intestinal radioactivity, while in one Group 2 patient this was not found. We observed two types of lymphoscintigraphic pattern in patients with intestinal lymphangiectasia. To clarify the exact pathophysiology, further study is required.

Adolescent↗
PubMed1984

Intestinal lymphangiectasia--a report in two Chinese children.

Two Chinese infants with asymmetrical lymphoedema and steatorrhoea were investigated. The laboratory investigations showed that they had hypoproteinaemia without proteinuria. One of them showed peripheral lymphopenia and hypogammaglobulinaemia. Radiologically, they had evidence of extensive lymphangiectasia of the small intestine. This was confirmed histologically. Endoscopy of one of them showed diffuse milky nodular duodenal mucosa. The appearance of the mucosa was rather typical of intestinal lymphangiectasia.

Endoscopy↗
PubMedDate not supplied

Ultrasonographic diagnosis--small bowel lymphangiectasia in a dog.

A 7-year-old, West Highland White Terrier had a 5-month history of diarrhea, dysorexia, and weight loss. Sonographically, there was a focal area of intestinal thickening with loss of layering. A neoplastic or severe inflammatory condition was suspected and intestinal lymphangiectasia was diagnosed histopathologically. This patient seems to be the first description of intestinal lymphangiectasia appearing as an intestinal mass.

Animals↗
PubMed1992

Coeliac disease and lymphangiectasia.

Two out of 74 children with coeliac disease demonstrated severe intestinal protein loss. In both children a serial small bowel biopsy specimen showed intestinal lymphangiectasia to be also present. Intestinal lymphangiectasia is another disorder that may be associated with coeliac disease.

Celiac Disease↗
PubMed2000

Very late onset small intestinal B cell lymphoma associated with primary intestinal lymphangiectasia and diffuse cutaneous warts.

As only a handful of lymphoma cases have been reported in conjunction with primary intestinal lymphangiectasia, it is not yet clear if this association is merely fortuitous or related to primary intestinal lymphangiectasia induced immune deficiency. We report on two female patients, 50 and 58 years old, who developed small intestinal high grade B cell lymphoma a long time (45 and 40 years, respectively) after the initial clinical manifestations of primary intestinal lymphangiectasia. They presented with a longstanding history of fluctuating protein losing enteropathy, multiple cutaneous plane warts, and markedly dilated mucosal and submucosal lymphatic channels in duodenal biopsies. One had a large ulcerated tumour of the proximal ileum and the other diffuse ileal infiltration. In both, histological examination showed centroblastic high grade B cell lymphoma associated with duodenojejuno-ileal mucosal and submucosal lymphangiectasia. They were subsequently successfully treated with surgery and postoperative chemotherapy (AVmCP: adriamycin, cyclophosphamide, Vm26, and prednisolone), and chemotherapy alone (PACOB: adriamycin, cyclophosphamide, vincristine, bleomycine, and prednisolone), respectively. A three year follow up in both cases showed persistent diffuse lymphangiectasia without evidence of lymphoma. The present findings support the hypothesis that primary intestinal lymphangiectasia is associated with lymphoma development.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1981

Primary intestinal lymphangiectasia. Case report with radiological and ultrastructural study.

A 16-year-old boy with primary intestinal lymphangiectasia presented with peripheral edema of 6 weeks duration. Laboratory and radiological studies included absolute lymphopenia, hypoalbuminemia, steatorrhea, abnormal stool 51Cr-albumin value, edema of small intestinal folds, dilated mesenteric lymphatics, as well as bilateral filling defects in external and common iliac lymph nodes. Abdominal CT scan revealed a possible periaortic mass. Small intestinal biopsies demonstrated normal villous architecture with dilated lacteals, and electron microscopy revealed enterocytes with normal as well as blunted microville, enlarged, dilated lacteals and intercellular vacuoles. An exploratory laparatomy was performed and revealed no masses but dilated serosal lymphatic vessels. Medical treatment, including marked restriction of long chain triglyceride intake, resulted in loss of peripheral edema, weight stabilization, and normal activity.

Adolescent↗
PubMed1976

Lymphangiographic findings in Behçet's disease with lymphangiectasia of the small intestine.

Bipedal lymphography was performed in 5 cases of Behçet's disease. In 4 of 5 cases, histologic examination of intestinal biopsy specimens showed lymphatic dilatation of the small intestine which were considered to be a characteristic finding of intestinal lymphangiectasia, a protein-losing enteropathy, but no patient with Behçet's disease showed reduced concentration of serum total protein as well as abnormal value of 131I-PVP test. Lymphograms showed an increase in number of iliopelvic and lumbar lymphatic vessels as a major finding, but they failed to demonstrate a hypoplasia or aplasia of lymphatic system or obstruction of thoracic duct. It is conceivable that the lymphatic dilatation of the small intestine in Behçet's disease may be related to increased flow of lymph due to excessive vascular hyperpermeability and may not be related to a block of lymphatic system which has been considered to be a cause of enteric protein loss in intestinal lymphangiectasia. The authors wish to emphasize that the dilatation of lymphatic vessel of the small intestine is not a sufficient finding to indicate the presence of protein-losing enteropathy.

Adult↗
PubMedDate not supplied

Intestinal crypt lesions associated with protein-losing enteropathy in the dog.

Six dogs were diagnosed with protein losing enteropathy (PLE). There was no evidence of inappropriate inflammatory infiltrates or lymphangiectasia in multiple mucosal biopsies of the small intestine of 4 of the dogs. The 5th and 6th dogs had obvious lymphangiectasia and a moderate infiltrate of inflammatory cells in the intestinal mucosa. All 6 dogs had a large number of dilated intestinal crypts that were filled with mucus, sloughed epithelial cells, and/or inflammatory cells. Whether PLE occurs in these dogs because of protein lost from the dilated crypts into the intestinal lumen or whether the dilated crypts are a mucosal reaction due to another undetermined lesion that is responsible for alimentary tract protein loss is unknown. However, when large numbers of dilated intestinal crypts are present, they appear to be associated with PLE even if there are no other remarkable lesions in the intestinal mucosa.

Animals↗
PubMed1976

Pathophysiology of enlargement of the small bowel fold.

Enlargement of the valvulae conniventes is an integral part of the pattern diagnosis of primary small bowel disease causing malabsorption. The pathophysiology underlying enlargement of the fold and the most typical diseases with prominent folds leading to malabsorption are discussed. Differential diagnosis is based on enlargement of the fold and on secondary signs.

Amyloidosis↗
PubMed2001

Clinical, clinicopathologic, radiographic, and ultrasonographic characteristics of intestinal lymphangiectasia in dogs: 17 cases (1996-1998).

OBJECTIVE: To characterize the clinical, clinicopathologic, and imaging findings in dogs with intestinal lymphangiectasia and to compare the histologic grade of lymphangiectasia with clinicopathologic and imaging abnormalities. DESIGN: Retrospective study. ANIMALS: 17 dogs with a histologic diagnosis of intestinal lymphangiectasia. PROCEDURE: Medical records of dogs with a histologic diagnosis of intestinal lymphangiectasia were reviewed for signalment, history, clinical signs, results of exploratory laparotomy, and clinicopathologic, radiographic, ultrasonographic, and histologic findings. RESULTS: Mean age of dogs was 8.3 years; the most common clinical signs were diarrhea, anorexia, lethargy, vomiting, and weight loss. Abnormal physical examination findings included dehydration, ascites, and signs of pain on palpation of the abdomen. The most notable clinicopathologic findings were low serum ionized calcium concentration and hypoalbuminemia. Abdominal ultrasonography was performed in 12 dogs and revealed intestinal abnormalities in 8 dogs and peritoneal effusion in 7 dogs. Exploratory laparotomy revealed abnormalities in 9 of 16 dogs including thickened small intestine, dilated lacteals, lymphadenopathy, and adhesions. On histologic examination of the small intestine, concurrent inflammation was observed in 15 of 17 dogs, crypt ectasia in 5 of 17, and lipogranulomas in 2 of 17. CONCLUSIONS AND CLINICAL RELEVANCE: Intestinal lymphangiectasia in dogs appears to be a heterogeneous disorder characterized by various degrees of panhypoproteinemia, hypocholesterolemia, lymphocytopenia, and imaging abnormalities. In most dogs, the severity of hypoalbuminemia appears to offer the best correlation with severity of histologic lesions of lymphangiectasia. Imaging abnormalities are common in dogs with intestinal lymphangiectasia but are not specific enough to differentiate this disorder from other gastrointestinal disorders, nor are they predictive of histologic severity.

Abdomen↗
PubMed1980

Endogenous faecal calcium in chronic malabsorption syndromes and in intestinal lymphangiectasia.

Endogenous faecal calcium was measured by an isotopic technique in five patients with protein-losing enteropathy due to intestinal lymphangiectasia and in ten patients with chronic malabsorption due to severe Crohn's disease (one patient) or extensive small-bowel resection (nine patients). In most patients absorption of dietary calcium and calcium balance were also determined. Endogenous faecal calcium and digestive juice calcium were highly increased in 3 patients with intestinal lymphangiectasia and normal or subnormal in the remaining 12 patients. Absorption of dietary calcium was normal in patients with intestinal lymphangiectasia but extremely low in most patients with chronic malabsorption syndromes. It is concluded that a net loss of calcium in stools in patients with intestinal lymphangiectasia is due to increased endogenous faecal calcium. In contrast, a net loss of calcium in stools in patients with extensive small-bowel resection is due to decreased absorption of dietary calcium with normal or almost normal endogenous faecal calcium.

Adult↗
PubMed1993

A case of primary intestinal lymphangiectasia.

A 26-year-old male patient who had an 8 years history of recurrent peripheral edema with diarrhea and hypoproteinemia was evaluated. Endoscopic jejunal and ileal biopsy revealed markedly dilated mucosal lymph vessels with no evidence of inflammation. 99mTc-labeled human serum albumin (HSA) scintigraphy showed significant activity accumulating in the gastrointestinal tract to represent 99mTc-HSA leakage into the bowel lumen. A diagnosis of protein losing enteropathy and intestinal lymphangiectasia could be made. After treatment with a high protein and fat restricted diet, his symptoms subsided and the serum protein level was normalized.

Adult↗