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At least 127 records · Page 7Linked to original sources
PubMed2006

Rare instance of gingival enlargement in Klippel-Trenaunay syndrome: a case report.

Klippel-Trenaunay Syndrome is a rare congenital malformation that may include port-wine stain, soft tissue and bony hypertrophy, and venous malformations and lymphatic abnormalities. Although it usually involves the limbs, it may also rarely involve the head, neck, and orofacial regions. Despite its rarity, Klippel-Trenaunay Syndrome should be considered in the differential diagnosis of gingival enlargement. The condition can be easily recognized clinically, but further investigations including imaging studies have to be carried out in order to better understand the nature of the lesion. This report describes a case of gingival enlargement in Klippel-Trenaunay Syndrome in a 16-year-old female patient. The diagnosis of the condition was made based on the patient history, clinical and radiographic examination, computed tomography (CT), and angiogram.

Adolescent↗
PubMed1974

An unusual variant of Klippel-Trenaunay-Weber syndrome.

The association of soft tissue and bony overgrowth, varicose veins and port-wine nevus is known as Klippel-Trenaunay-Weber syndrome. An unusual case of a young woman with this syndrome is presented. In addition to the classic features she had face swelling, bilateral limb involvement and an infected cutaneous lymphangioma. No evidence of arteriovenous fistula was demonstrated. In contrast to an abnormal lymphatic system the deep veins of the limbs were normal. Unilateral lung lesions were demonstrated which have not been described previously in such cases.

Adult↗
PubMedDate not supplied

Accidents in the surgical treatment of varicose veins.

Serious accidents in the surgical treatment of varices are quite exceptional and it is impossible to say with any accuracy how often they occur. Generally they result from major errors of surgical technique, and arise mainly from: haemorrhaging after the tearing of the femoral vein or because of the loosening of the stump of the saphenofemoral junction, traumatism of the femoral artery, damage of the nervus peronaeus in crossectomy of the saphena parva. Minor accidents are more frequent but here too it is impossible to state a precise percentage. There are four fairly common types of accident: serious haematoma following a stripping, which can be avoided by ligation of the accessible collaterals. Trendelenburg's peroperative position, aspiration drainage, and elastic compression; phlebitis, usually superficial and at the level of appendage clusters on the trunk of the saphenal vein, but sometimes deep: much rarer, this has not been observed once in 2335 cases of surgery of the long saphenous vein, and only three times in 583 cases of surgery performed either on the short saphenous vein alone, or on both saphenae; oedema of lymphatic origin to be observed particularly after operations on recurrent cases but sometimes after initial surgery too: in the case of pre-existing lymphatic abnormality or in the case of multiple incisions, especially internal crural incisions; painful paresthesia of saphenal nerves which, as a general rule, regress spontaneously.

Femoral Artery↗
PubMed1997

Management of chylothorax.

BACKGROUND: Chylothorax is a rare primary or secondary condition the optimum management of which remains uncertain. METHODS: Twenty cases of chylothorax, including ten of primary chylothorax and ten secondary to either malignancy, subclavian vein thrombosis or lymphangioma treated between 1956 and 1986 have been reviewed. RESULTS: Open pleurectomy was the most successful treatment in preventing reaccumulation of the effusion. Three patients had thoracic duct-azygous vein anastomoses, but all anastomoses were probably occluded within a year of surgery. Three patients have been lost to follow-up and five died within 2 years of their treatment, but 12 patients were alive and free from an effusion 3-22 years after treatment. CONCLUSION: Patients with chylothorax should undergo lymphangiography to identify the cause and site of the lymphatic abnormality. Conservative treatment is successful in some patients but should be abandoned if the fluid loss exceeds 1.5 l/day for more than 5-7 days in an adult or more than 100 ml/day in a child. Parietal pleurectomy is the most successful treatment when no distinct chylous leak can be identified. Less commonly, an isolated chylous leak either in the chest or in the abdomen may be identified and this should be treated by direct ligation.

Adult↗
PubMed1998

Octreotide in the treatment of intestinal lymphangiectasia.

Primary intestinal lymphangiectasia is characterized by dilated small bowel lymphatics and loss of lymph into the bowel lumen resulting in hypoproteinaemia and oedema. Some patients have a more generalized lymphatic abnormality associated with lymphoedema of the limbs and chylous pleural effusions. There is no specific treatment although enteric protein loss may decrease with a low-fat diet. This report describes a patient with severe primary intestinal lymphangiectasia, associated with limb oedema and recurrent pleural effusions, who responded to treatment with octreotide. Before starting octreotide she required weekly intravenous albumin infusions to maintain the serum albumin above 20 g/l. Bilateral pleural effusions repeatedly reaccumulated despite pleurectomy and subsequently tetracycline pleurodesis. Treatment with octreotide, 200 microg twice daily, resulted in a reduction in enteric protein loss from 16 to 4.1% in 5 days (normal less than 1%) and the serum albumin was maintained between 22 and 26 g/l without the need for albumin infusion. Oedema in the arms resolved completely and the pleural effusions did not reaccumulate. The mechanism of action of octreotide in this condition appears to be due to a reduction in gut protein loss and another, as yet unidentified, action.

Adolescent↗
PubMed1990

Nuchal cystic hygroma associated with massive lymphangiomatosis of the upper extremity and myocardium in a stillborn fetus: a case report.

Nuchal cystic hygromas may be encountered in stillborn fetuses as part of a spectrum of a more generalized abnormality of the lymphatic system. These lymphatic system abnormalities may be accompanied by various visceral anomalies, especially in Ullrich-Turner Syndrome (UTS) (45,X0) but also in non-45,XO fetuses. We present unique autopsy findings in the case of a stillborn female fetus with a nuchal cystic hygroma associated with massive lymphangiomatosis of the upper extremity as well as myocardium. While previous descriptions of abnormal cardiac lymphatics associated with nuchal cystic hygroma have focused on dilated lymphatics surrounding the root of the aorta and pulmonary artery, the present case extends the spectrum of possible findings to include subtotal replacement of the ventricular myocardium by dilated lymphatics.

Cardiomegaly↗
PubMed2004

Defective valves and abnormal mural cell recruitment underlie lymphatic vascular failure in lymphedema distichiasis.

Lymphatic vessels are essential for the removal of interstitial fluid and prevention of tissue edema. Lymphatic capillaries lack associated mural cells, and collecting lymphatic vessels have valves, which prevent lymph backflow. In lymphedema-distichiasis (LD), lymphatic vessel function fails because of mutations affecting the forkhead transcription factor FOXC2. We report that Foxc2(-/-) mice show abnormal lymphatic vascular patterning, increased pericyte investment of lymphatic vessels, agenesis of valves and lymphatic dysfunction. In addition, an abnormally large proportion of skin lymphatic vessels was covered with smooth muscle cells in individuals with LD and in mice heterozygous for Foxc2 and for the gene encoding lymphatic endothelial receptor, Vegfr3 (also known as Flt4). Our data show that Foxc2 is essential for the morphogenesis of lymphatic valves and the establishment of a pericyte-free lymphatic capillary network and that it cooperates with Vegfr3 in the latter process. Our results indicate that an abnormal interaction between the lymphatic endothelial cells and pericytes, as well as valve defects, underlie the pathogenesis of LD.

Animals↗
PubMed1996

Lymphatic function in the yellow nail syndrome.

Peripheral oedema is commonly seen in the yellow nail syndrome (YNS). Contrast lymphangiography has shown abnormal collecting lymphatics in some patients with YNS. In this study, lymphatic function in the upper and lower limbs of 17 patients with YNS, in normal controls, and in patients with established classical lymphoedema, has been assessed using quantitative lymphoscintigraphy. Nine subjects with YNS had swelling of the legs and two had features typical of lymphoedema. The lymphatic drainage was significantly reduced in the legs of patients with YNS but not to the level seen in lymphoedema. Lymphatic function was also reduced in the arms in patients with YNS. Venous insufficiency did not contribute to the leg oedema. These results suggest that the underlying cause of YNS is not primarily a lymphatic abnormality. The lymphatic impairment associated with YNS appears to be secondary, and predominantly functional in nature, rather than due to structural changes.

Adolescent↗
PubMed2000

[Vascular and neural malformations].

The presentation of vascular and neural malformations in an understandable terminology permits accurate diagnosis, proper treatment, individualized prognosis, and also stimulates studies of pathogenesis. The descriptive classification includes: NF 1 and NF 2; hemangiomas, low- and high-flow vascular malformations, combined malformations, and hypertrophy; and syndromes such as, Parkes Weber, Klippel-Trénaunay, Maffuci's, and multiple dysplasia syndromes. Lymphatic malformations are abnormalities of lymphatic development. The list of treatment includes surgical and nonsurgical treatment. Not all vascular malformations can be successfully treated. Coping with NF is a challenge for both, affected individuals and health care professionals. NF is often associated with a myriad of anomalies that present a lot of problems for plastic surgery. In certain cases watchful waiting seems justified but not in cases of severe problems, giant growth, and local complications.

Adolescent↗
PubMed2006

Onset of abnormal blood and lymphatic vessel function and interstitial hypertension in early stages of carcinogenesis.

Recent improvements in diagnostic methods have opened avenues for detection and treatment of (pre)malignant lesions at early stages. However, due to the lack of spontaneous tumor models that both mimic human carcinogenesis and allow direct optical imaging of the vasculature, little is known about the function of blood and lymphatic vessels during the early stages of cancer development. Here, we used a spontaneous carcinogenesis model in the skin of DNA polymerase eta-deficient mice and found that interstitial fluid pressure was already elevated in the hyperplastic/dysplastic stage. This was accompanied by angiogenic blood vasculature that exhibited altered permeability, vessel compression, and decreased alpha-smooth muscle actin-positive perivascular cell coverage. In addition, the lymphatic vessels in hyperplastic/dysplastic lesions were partly compressed and nonfunctional. These novel insights may aid early detection and treatment strategies for cancer.

Animals↗
PubMedDate not supplied

Abnormalities of the leg lymphatics are not specific for bancroftian filariasis.

Studies using conventional angiography or non-invasive scintigraphy have revealed widespread abnormalities in the lymphatics of the legs of patients with bancroftian filariasis, regardless of whether clinical lymphoedema is present. To determine if the observed changes were specific for filarial infections, we imaged the lymphatics of both legs in native residents of an area in Brazil where filariasis is not endemic. Study participants were matched by age, socioeconomic status and physical activities to patients with filariasis in Recife, evaluated in parallel. Based on textbook criteria, only one of 15 study participants had a completely normal lymphoscintigram. Modest to severe pathology of the leg lymphatics was observed in the remaining 14 residents of the non-endemic area and in 49 of 50 patients with bancroftian filariasis. These results indicated that factors other than filarial worms are a common cause of subclinical pathology of the leg lymphatics in north-eastern Brazil, and that the latter is not specific for bancroftian filariasis.

Brazil↗