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PubMed1976

Macrocheilia due to lymphangiectasia.

A case of symmetrical, diffuse macrocheilia due to lymphangiectasia is presented. No primary cause for this case or for many other pathologic forms of macrocheilia has been established. Tranverse mucosal wedge resection of the lip with mucosal advancement, however, is a satisfactory method of treatment.

Adolescent↗
PubMed1979

Cystic lymphangioma of the spleen.

Splenic cysts are uncommon, with fewer than 600 cases reported in the world literature. This report concerns a case of cystic lymphangioma of the spleen, consisting of a large splenic cyst with associated multiple, small, subcapsular cysts and lymphangiectasia. The cysts are lined by endothelium, and they conform to the characteristics of dilated lymphatic channels. Lymphangioma of the spleen is thought to arise from congenital obstruction of lymphatic flow, with subsequent dilatation of lymphatic channels. The diagnosis and treatment of splenic cysts are discussed and a new classification of splenic cysts is proposed.

Adult↗
PubMed2000

Two brothers with Hennekam syndrome and cerebral abnormalities.

We report two brothers with mental retardation, lymphoedema of the limbs and facial anomalies. Hennekam et al. (Am J Med Genet 34:593-600; 1989) described four patients with identical signs and intestinal lymphangiectasia. To confirm the diagnosis of Hennekam syndrome we undertook a duodenal biopsy from the older brother which revealed intestinal lymphangiectasia. So far only one patient with Hennekam syndrome and cerebral abnormalities has been described. This patient presented with pachygyria in the parietal area. Cerebral MRI in our two cases revealed small subcortical hyperintensities in both patients and a large cystic lesion in the younger patient probably representing an old media infarction.

Abnormalities, Multiple↗
PubMed2003

Further delineation of Hennekam syndrome.

We report four children from four inbred Arab families with varying manifestations of Hennekam syndrome and additional features that have not been previously reported. These include abnormalities of the middle ear, anomalous pulmonary venous drainage, interrupted inferior vena cava, polysplenia, crossed renal ectopia, median position of the liver and multiple cavernous haemangiomas. In addition, in one case lymphoedema was absent and oedema due to hypoproteinaemia appeared at 6 years of age. Since anomalies of the veins and the consequent developmental abnormalities of the lymphatics might lead to alterations in the fluid balance of the embryo, we hypothesize that altered fluid dynamics due to defective vascular and lymphatic development might disrupt critical events in craniofacial morphogenesis resulting in Hennekam syndrome.

Abnormalities, Multiple↗
PubMed1999

A case of intra-abdominal multiple lymphangiomas in an adult in whom the immunological evaluation supported the diagnosis.

A 60-year-old patient with intra-abdominal lymphangiomatosis is described. He presented with anaemia due to enteric haemorrhage, hypoproteinaemia with heavy hypogammaglobulinaemia and T-cell lymphopenia. Duodenal biopsy showed lymphangiectasia while a small bowel study revealed several filling defects in the terminal ileum. On exploratory laparotomy, numerous inoperable lymphangio-haemangiomata were found, involving the small and large intestine, appendix, mesenterium, gallbladder and main biliary tract. The importance of T-cell lymphopenia and hypogammaglobulinaemia in the diagnosis of intra-abdominal lymphangiomatosis with lymphangiectasia is stressed.

Abdominal Neoplasms↗
PubMed2005

Remission of protein-losing enteropathy after nodal lymphoma treatment in a patient with primary intestinal lymphangiectasia.

Primary intestinal lymphangiectasia (PIL), so-called Waldmann's disease, is an uncommon condition, characterized by dilated intestinal submucosal and subserosal lymphatics of the gastrointestinal tract. Protein-losing enteropathy is the most common manifestation of this supposed congenital disease. Since the initial description in 1961, 11 cases of lymphoma have been reported suggesting that PIL predisposes to lymphoma. Here, we report the first case of primary nodal location lymphoma during PIL with recovery of the protein-losing enteropathy after its treatment by radiochemotherapy.

Adult↗
PubMed2006

Acquired intestinal lymphangiectasia successfully treated with a low-fat and medium-chain triacylglycerol-enriched diet in a patient with liver transplantation.

Intestinal lymphangiectasia is defined as a dilatation of small bowel lymphatic capillaries and a loss of lymph into the bowel lumen. Clinically it is characterized by hypoproteinaemia and oedema. We present here a case of protein-losing enteropathy due to intestinal lymphangiectasia after liver transplantation in a 57-year-old man who was transplanted for hepatitis C virus. Four years after liver transplantation, the patient developed hypoalbuminaemia and ascites associated with recurrence of cirrhosis. The sudden fall in serum albumin led us to look for a cause of reduction other than or in addition to cirrhosis. Duodenal biopsies showed tall villi with dilated lymphatic vessels and widening of the villi caused by oedema, demonstrating intestinal lymphangiectasia. In this case a low-fat diet supplemented with medium-chain triacylglycerols achieved an early clinical improvement with increased serum albumin levels and ascites disappearance. Intestinal lymphangiectasia should be suspected in liver-transplanted patients developing hypoproteinaemia and hypoalbuminaemia after the recurrence of cirrhosis.

Diet, Fat-Restricted↗
PubMed1997

'Swiss cheese' lymphangiectasia in a case of solitary morphea profunda.

Reports of lymphangiectases that occur in lesions of morphea are rare. We describe a 24-year-old woman with a solitary morphea profunda associated with lymphangiectasia. Unlike previously reported cases, our case showed lymphatic dilatation resembling Swiss cheese and developed around a milium.

Adult↗
PubMed1993

Acquired cutaneous lymphangiectasia.

We report a case of acquired cutaneous lymphangiectasia associated with underlying malignancy, and following an arthrotomy. Clinical and histological features were identical with those of acquired lymphangioma. Lymphangiography showed signs of acquired lymphatic obstruction. Both the underlying tumour and the arthrotomy were thought to be contributory factors.

Adult↗