Cases reported "Lymphangiectasis"

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11/53. drainage of subcutaneous lymphatic fluid for the management of respiratory distress in a case of generalized lymphangiectasia in an infant.

    A 10-month-old girl was referred to our hospital because of congenital and persistent bilateral chylothorax and generalized lymphedema as well as long-standing respiratory disturbance. Radiological studies showed a diffuse network of superficial lymphatic vessels without major trunks throughout her entire body as well as the lung. She was diagnosed with systemic lymphangiomatosis complicated with pulmonary lymphangiectasia. Percutaneous puncture in the lower leg was performed to discharge the lymphatic fluid and proved to be effective for the respiratory disturbance. This procedure is safe and easy and effectively improves the quality of life of the patient and the family in case of such a persistent disease.
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12/53. Pulmonary abnormalities in Klippel-Trenaunay syndrome. A histologic, ultrastructural, and immunocytochemical study.

    Klippel-Trenaunay (KT) syndrome is a rare, sporadic, congenital vascular disease of unknown etiology. We describe pulmonary findings in an 18-year-old male patient followed up since birth with the KT syndrome. The patient developed pleural and pericardial serous effusions that led to an open lung biopsy. Previous pulmonary findings have been limited to thromboembolic phenomena and pulmonary vein varicosities. On the other hand, reports of lymphatic hyperplasia, aplasia, and hypoplasia in KT have been limited to the extremities. For the first time, we describe lymphatic involvement of the lung in KT. The plexiform hyperplasia of the lymphatic channels with smooth muscle hyperplasia leading to lymphatic obstruction, pleural and pericardial effusions are new findings. The lymphatic nature of the plexiform channels was confirmed by immunohistochemistry. von willebrand factor and QD-END/10 monoclonal antibodies either did not react or reacted poorly with lymphatic endothelium, features used to distinguish lymphatic and venous endothelium. Ultrastructurally, the absence of basement membrane continuity further substantiated the lymphatic nature of the channels. From our findings, the lymphatic abnormality in the syndrome appears to be more generalized than previously thought. This entity should be distinguished from lymphangioleiomyomatosis to which it bears a superficial morphologic appearance.
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13/53. Surgical management of conjunctival lymphangiectasis by conjunctival resection.

    PURPOSE: To report the treatment of symptomatic conjunctival lymphangiectasis with conjunctival resection. DESIGN: Interventional case series. methods: Retrospective review of three cases of patients with symptomatic unilateral conjunctival lymphangiectasis who had conjunctival resection of the involved tissue down to bare sclera. RESULTS: The surgical area reepithelialized without recurrence of the lymphatics. In all cases the patients have remained asymptomatic for at least 14 months. CONCLUSION: Conjunctival resection for symptomatic lymphangiectasis can be a successful therapeutic option for this condition.
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14/53. 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.
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15/53. Congenital pulmonary lymphangiectasia: CT and pathologic findings.

    Congenital pulmonary lymphangiectasia is a rare disease characterized by dilation of lymphatic channels without lymphatic proliferation. The disease is seen almost exclusively in infancy and early childhood. The authors report 2 cases of pulmonary lymphangiectasia. The patients were a 12- and a 25-year-old male who presented with progressive dyspnea and hemoptysis. The diagnosis was confirmed by open lung biopsy. The radiographic findings consisted of bilateral reticular changes, peribronchial cuffing, and bilateral pleural effusions. High-resolution CT demonstrated extensive bilateral septal and peribronchovascular interstitial thickening, areas of ground-glass attenuation, and bilateral pleural effusions. The histologic findings consisted of ectatic and tortuous lymphatic channels in the interlobular septa, bronchovascular sheaths, and pleura.
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16/53. autopsy case of congenital pulmonary lymphangiectasis.

    Congenital pulmonary lymphangiectasis (CPL) is a rare anomaly. We report a female infant born at 39 weeks of gestation who was found to have CPL. cyanosis and tachypnea were noted immediately after birth, and, at room air, PaO2 was 30.7 mmHg, PaCO2 was 82.5 mmHg and pH was 7.12. The infant's symptoms did not improve even with the initiation of artificial ventilation. Chest x-ray film showed cotton-like infiltrates in both lungs and an air-leak surrounding the cardiac shadow. echocardiography study showed no abnormality. The neonate died 3 days after birth due to hypoxemic cardiac failure. At autopsy, the pleural surface contained numerous dilated vessels that had the appearance of lymphatics. Microscopic features of the lungs were marked lymphatic dilatation of the perivascular, subpleural and interlobular areas. lymphangiectasis was found in the liver, kidney, pancreas, thyroid and alimentary canals, such as the esophagus, stomach and rectum. patients with lymphatic dilatations in extrapulmonary organs have mild pulmonary involvement and symptoms and a better prognosis. However, a few cases of CPL with lymphatic dilatations in extrapulmonary organs and an aggressive course, such as the present case, have been reported. The clinical behavior and prognosis of CPL depend on the extent of pulmonary involvement of the lymphatic dilatations regardless of systemic lymphatic dilations.
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17/53. Acquired lymphangiectasis after breast conservation treatment for breast cancer: report of a case.

    Acquired lymphangiectasis is a dilatation of lymphatic vessels that can result as a complication of surgical intervention and radiation therapy for malignancy. Acquired lymphangiectasis shares clinical and histological features with the congenital lesion, lymphangioma circumscriptum. diagnosis and treatment of these vesiculobullous lesions is important because they may be associated with pain, chronic drainage, and cellulitis. We describe patient who had these lesions after treatment for cancer. Although a number of treatment options are available, we have found cryosurgery and electrocautery to be particularly effective.
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keywords = lymphatic
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18/53. Diffuse pulmonary lymphangiectasis with heart defect discovered 4 months post-natally.

    Congenital pulmonary lymphangiectasia is a rare abnormality with dilatation of pulmonary lymphatics and the radiological pattern of a pulmonary interstitial syndrome. It is usually symptomatic at birth and is almost always lethal. We report the case of an infant with congenital pulmonary lymphangiectasis and congenital heart disease who is still alive at 11 years. This case is interesting from the pathogenic, nosologic and prognostic point of view.
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keywords = lymphatic
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19/53. Lipedematous scalp and lipedematous alopecia: a clinical and histologic analysis of 3 cases.

    BACKGROUND: Lipedematous alopecia and lipedematous scalp are two similar unusual conditions mostly affecting healthy black women. OBJECTIVE: The purpose of our study was to report three cases of this condition with emphasis on clinical and histologic findings, and to review the literature on the subject. methods: The study includes clinical, echographic, and histologic findings of 3 patients, complemented with a literature review. RESULTS: Two cases of lipedematous alopecia and one of lipedematous scalp in 3 white women had echographic confirmation of an increased subcutaneous layer. The presence of ectatic lymphatic vessels in the two cases with hair loss was particularly emphasized. CONCLUSIONS: Our findings suggest a lessened role of racial factors, but confirms the sex implications in these related conditions, and stress the potential significance of lymphangiectatic vessels in the development of alopecia in these patients.
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keywords = lymphatic
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20/53. Pulmonary lymphangiectasis in an asymptomatic adult.

    Pulmonary lymphangiectasis is a rare anomaly in infancy that is characterized by dilatation of pulmonary lymphatic vessels resulting in fetal respiratory distress. Pulmonary lymphangiectasis is considered to occur exclusively in young children and neonates, and very few survive beyond an early age. We herein present an asymptomatic adult case of localized pulmonary lymphangiectasis with multiple nodules. A 27-year-old asymptomatic female presented with multiple nodules on chest computed tomogram images. An exploratory video-assisted thoracoscopy revealed multiple yellowish cysts on the visceral pleura, which were histologically diagnosed as lymphangiectasis. In the present study, the pathogenesis and clinical characteristics are discussed.
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