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At least 19 recordsLinked to original sources
PubMed1979

Lymphatic abnormalities in Noonan syndrome: A case report.

Lymphatic abnormalities are not generally recognized as part of the Noonan syndrome. A child with this condition in whom unique and widespread lymphatic abnormalities were demonstrated by lymphography is described. Both T and B lymphocytes were detected in chylous fluid drained from the thorax. In addition, the child was found to have a protein-losing enteropathy and cardiovascular defects. The clinical spectrum of the Noonan syndrome may include animalies of the lymphatic system.

Cardiac Catheterization↗
PubMed1978

Lymphatic abnormalities in Noonan's syndrome.

Five boys who had Noonan's syndrome and lymphatic abnormalities are reported. The youngest boy had clinical lymphoedema and the other four showed dermal backflow after interdigital injection of Patent Blue indicating impairment of flow along the superficial lymphatics. One boy had severe bilateral chylothorax. The lymphographic findings in four of these boys are reported. Patients with the Noonan syndrome frequently have oedema of the hands and feet at birth, which decreases during the first years of life [10]. It has been demonstrated by lymphography that similar peripheral oedema in patients with the Turner's syndrome is due to lymphatic hypoplasia [1, 3]. We report certain lymphatic abnormalities diagnosed by lymphography in four out of five patients with Noonan's syndrome.

Adolescent↗
PubMed1992

Spinal cord arteriovenous malformation in a person with congenital lymphatic abnormalities.

Spinal cord arteriovenous malformations have been described in association with a variety of congenital diseases affecting the vasculature, including Klippel-Trenaunay-Weber syndrome, Rendu-Osler-Weber syndrome and others, but rarely in association with lymphatic abnormalities. We report the case of a young man with congenital lymphedema and arteriovenous malformations of one lower extremity and a spinal cord arteriovenous malformation. Awareness of the possible presence of a central nervous system arteriovenous malformation in individuals with pre-existing arteriovenous and lymphatic abnormalities may be helpful in their diagnosis and management.

Abnormalities, Multiple↗
PubMed1999

Bronchial casts in children with cardiopathies: the role of pulmonary lymphatic abnormalities.

Expectoration of bronchial casts, a condition also called plastic bronchitis, is very rare in children. Bronchial casts may be associated with bronchopulmonary diseases associated with mucus hypersecretion, bronchopulmonary bacterial infections, congenital and acquired cardiopathies, or pulmonary lymphatic abnormalities. A classification based on anatomy and pathology has been proposed which identifies an "acellular" group associated with congenital cardiopathies and palliative surgery. We report on 3 cases with bronchial casts associated with cardiopathy. Observations suggest that the formation of bronchial casts may result from lymphatic leakage into the bronchi. The 3 cases on which we report were immunodeficient and had pulmonary lymphatic abnormalities. The bronchial casts contained lymphocytes and lipids, as determined by histologic examination. In the absence of congenital pulmonary or diffuse lymphatic dysplasia associated with cardiopathy, the principal factors resulting in the formation of bronchial casts appear to be surgical trauma to the lymphatic channels surrounding the bronchi, pleural adhesions, and high systemic venous blood pressure. The prognosis for these patients is poor, and possibilities for treatment are limited.

Bronchial Diseases↗
PubMed1981

Lymphatic abnormalities in protein-losing gastropathy, especially in Ménétrier's disease.

Histopathologic and lymphographic studies were performed in 4 patients with protein-losing gastropathy to elucidate the degree of lymphatic disorders and their contribution to clinical features. Histologic examination of gastric mucosa showed various degrees of hyperplasia of the surface epithelium and cystic dilatation of glands, which were considered to be characteristic findings of protein losing gastropathy. Moreover, in 3 of 4 patients there were edematous change of the lamina propria or the submucosa, and marked lymphangiectasia was seen in the submucosa of 3 patients and in the lamina propria of the other. Performed lymphograms showed an increase in the number of lymphatic vessels and lymph nodes in the iliopelvic and lumbar regions of 2 of the patients. This high incidence of lymphatic abnormalities in patients with protein-losing gastropathy suggested that such systemic lymphatic disorders were an important etiologic factor. However, such abnormalities in lymphatic system were not demonstrated in all cases of protein-losing gastropathy. Therefore, lymphatic disorders were not considered to be the primary cause of gastric protein loss, but the important factor that modifies the variety of lost protein and its clinical features.

Adult↗
PubMed1989

Lymphocyte subpopulations in children with abnormal lymphatic circulation.

Peripheral blood lymphocyte subsets were enumerated in five children with abnormal lymphatic circulation (three with lymphangiectasia, one with chylothorax, and one child with chyloperitoneum). All patients were lymphopenic. The percentage and absolute number of blood T-lymphocytes (CD3) were low in two children and normal in the other children. The percentage and absolute number of helper/inducer lymphocytes (CD4) were markedly reduced in all patients. The percentage of suppressor/cytotoxic lymphocytes (CD8) was normal or elevated in all children, and the absolute number of CD8 cells was normal in three patients. The CD4/CD8 ratio was reversed in all patients. In the two patients tested, the proliferative response of peripheral blood mononuclear cells to mitogens was reduced. T-lymphocyte subsets were measured in the pleural or peritoneal fluid of three patients, and the CD4/CD8 ratio was normal or increased. In each child, the CD4/CD8 ratio in the lymphatic fluids was markedly higher than the CD4/CD8 ratio of the blood (4.0/0.45, 1.75/0.95, and 1.3/0.85). The reversed CD4/CD8 ratio in the blood in cases of chronic loss of chyle may be due to either selective transport of CD4 lymphocytes into the lymphatic fluids or a shorter half-life of CD8 compared to CD4 lymphocytes. This finding may in part explain the abnormal cellular immunity previously observed in patients with lymphangiectasia.

Adolescent↗
PubMed1991

Lymphatic abnormalities in Alagille's syndrome.

Chylous pleural effusions developed in a patient with Alagille's syndrome who had dysplasia of the lymphatic system. Lymphatic abnormalities are not a recognised feature of Alagille's syndrome.

Adult↗
PubMed1994

Lymphoscintigraphic analysis of lymphatic abnormalities in symptomatic and asymptomatic human filariasis.

To obtain high-resolution radionuclide lymphoscintigraphic images of affected limbs in persons with both symptomatic and asymptomatic filarial infection, 36 volunteers were recruited from a Wuchereria bancrofti-endemic area of Recife, Brazil, for a prospective, controlled analysis. Subjects were stratified after determination of serologic and clinical determinants of filarial infection status. Widespread lymphatic abnormalities were found in clinically asymptomatic microfilaremic persons, who had been assumed to have infection but not disease. All patients with clinical manifestations of lymphatic pathology and marked abnormalities. No correlation was found between clinical findings and actual lymphatic function as demonstrated by lymphoscintigraphy. The initial diagnosis of lymphatic filariasis, whether asymptomatic or symptomatic, is based on nonimaging laboratory criteria. After diagnosis, lymphoscintigraphy is a valuable tool for initial assessment of any lymphatic damage. Changes in strategies for therapeutic interventions in asymptomatic microfilaremic persons, who are not usually aggressively treated, may be warranted.

Adolescent↗
PubMed1977

Congenital mixed vascular deformities of the lower limb: the relevance of lymphatic abnormalities to their diagnosis and treatment.

Nineteen patients with vascular deformities of the lower extremity were studied and were classified into three groups based on their clinical and angiographic findings. 1) The Klippel or Venous Dysplasia Group (n = 8) demonstrated: cutaneous nevi of metameric distribution, congenital varices (many of which were anomalous by phlebography), hypertrophy of bone and other tissues, but no evidence of arterial involvement by arteriography. 2) Congenital Arterio-Venous Fistulae Group (n = 7) showed: cutaneous angiomata, hypertrophy of bone and soft tissues, but arteriographic findings of single or multiple arterio-venous fistulae. 3) Scattered Angiomata Group (n = 4) had single or multiple angiomata involving the lower limb in particular. Arteriography was normal, while phlbography revealed venous abnormalities. Lymphography was performed on all patients. Six of 8 of the Klippel group demonstrated aplastic or hypoplastic lymphatics by lymphography. By contrast, all 7 of the patients in the Congenital Arterio-Venous Fistulae Group showed in their lymphographic findings-three of 4 of these patients had aplasia or hypoplasia. Fourteen of the 19 patients complained of painful swelling as a major symptom. Lymphatic fistulae and lymphoceles developed in several patients.Four patients underwent reduction operations for lymphedema. Lymphatic abnormalities as demonstrated by lymphography play a significant role in mixed vascular deformities of the lower extremities.

Abnormalities, Multiple↗
PubMed1995

Abnormal lymphatic function in presymptomatic bancroftian filariasis.

Despite the common association of filarial infection with elephantiasis, the great majority of those infected are in fact clinically asymptomatic microfilariae carriers. The assumption has been that infection but not disease exists in these presymptomatic persons. In an area Brazil where Wuchereria bancrofti is endemic, flow studies done with dynamic radionuclide lymphoscintigraphy were used to compare 30 limbs from asymptomatic microfilaremic subjects with 16 control limbs. Geometric mean values for T1/2 (19.8 vs. 37.7 min; P < .001), appearance time (7.9 vs. 27.9 min; P < .001), percent uptake at the region of interest (0.67% vs. 0.14%; P < .001), and peak activity (62.6 vs. 2.6 cps; P < .001) each indicated an enhanced pattern of rapid, increased lymph flow in asymptomatic microfilaremic subjects. The abnormal lymphatic function in these subjects indicates that current passive intervention strategies may need to change if the debilitating sequelae of this parasitic infection are to be avoided.

Adolescent↗
PubMed2004

Abnormal lymphatic development in trisomy 16 mouse embryos precedes nuchal edema.

Ultrasound measurement of increased nuchal translucency is a method of risk assessment for heart malformations and trisomy 21 in human pregnancy. The developmental background of this nuchal edema is still not sufficiently understood. We have studied the process in trisomy 16 mice that show nuchal edema and heart malformations. We used trisomy 16 and wild-type (WT) embryos from embryonic day (E) 12.5 to E18.5. In WT embryos at E13, bilateral jugular lymphatic sacs are visible that share a lymphatic-venous membrane with the jugular vein. We could not in any case discern a valve between these vessels. At E14 in the TS16 embryos, the lymphatic sacs become enlarged showing abnormally thickened endothelium, specifically at the site of the membrane. In these embryos, severe edema develops in the nuchal region. There is a very close colocalisation of the nerves with the vascular structures. The start of reorganization of the jugular lymphatic sac to a lymph node is observed in both wild-type and TS16 but is diminished in the latter. In conclusion, abnormal size and structure of the jugular lymphatic sacs coincides with the development of nuchal edema. A disturbance of lymphangiogenesis might be the basis for increased nuchal translucency that is often observed in diseased human fetuses.

Animals↗
PubMed1993

Lymphatic abnormalities in human filariasis as depicted by lymphangioscintigraphy.

BACKGROUND: Investigation into filarial lymphedema has been hampered by the lack of a simple, safe, and easily repeated test to image the peripheral lymphatic system. Recent refinements in radionuclide lymphangioscintigraphy have established this noninvasive technique as the initial procedure of choice for visualizing lymphatics. Accordingly, we applied lymphangioscintigraphy to patients with filariasis and, for purposes of interpretation, compared the findings with those in patients with non-filarial lymphedema. METHODS: Thirty-three patients with classic symptoms or signs consistent with acute or chronic filariasis underwent lymphangioscintigraphy, and the findings were compared with those in five patients without lymphatic dysfunction and in 50 other patients with primary or secondary lymphedema without exposure to filariasis. RESULTS: As in patients with nonfilarial lymphedema, scintigraphic abnormalities in the 33 patients with filariasis included delayed or absent tracer transport of the radiotracer (25 patients), tortuous and bizarre deep lymphatics (seven patients), dermal diffusion (15 patients), retrograde tracer flow (six patients), and faint or absent regional nodal visualization (14 patients). Even in patients with long-standing filarial lymphedema, peripheral trunks were often visualized (at least in part), and regional nodes and more central lymphatics sometimes filled after light exercise. In some of the latter patients, however, discrete lymphatic trunks were not detected. CONCLUSION: Lymphangioscintigraphy is a simple, safe, reliable, noninvasive method with which to examine the peripheral lymphatic system, including truncal and nodal abnormalities, in endemic populations with occult and overt lymphatic filariasis.

Adolescent↗