Cases reported "Lymphedema"

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1/158. Lymphoedema, lymphatic microangiopathy and increased lymphatic and interstitial pressure in a patient with Parkinson's disease.

    New microvascular findings in a woman with Parkinson's disease and lower leg oedema are presented. Microlymphography showed enhanced filling of the microlymphatic network with the fluorescent contrast medium comparable to the findings in lymphoedema. The microlymphatic and interstitial pressures of the skin were increased (27 mm Hg and 16 mm Hg respectively). The increased interstitial and microlymphatic pressures are the result of insufficient venous and lymphatic drainage due to impairment of calf muscle function during walking in Parkinson's disease. Manual lymph drainage and compression therapy, in combination with improvement of calf muscle function, resulted in regression of the oedema.
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2/158. Autonomic regulation of lymphatic flow in the lower extremity demonstrated on lymphoscintigraphy in patients with reflex sympathetic dystrophy.

    PURPOSE: nuclear medicine techniques were used to show that the peripheral lymphatics are under autonomic control in much the same way as the blood vessels that supply the same anatomic region. methods: Three patients with complex regional pain syndrome type 1 (reflex sympathetic dystrophy) involving a lower extremity were evaluated using three-phase bone scintigraphy and peripheral lymphoscintigraphy. Each patient was treated with ipsilateral chemical lumbar sympathectomy, and lymphoscintigraphy was repeated within several days of the procedure. RESULTS: All three patients had evidence of decreased flow (compared with the contralateral extremity) to normal flow after ipsilateral sympathectomy. Bone scintigraphy, before and after sympathectomy, was difficult to interpret because of the effects of altered weight bearing. Two patients who had unilateral peripheral edema showed marked improvement after sympathectomy and increased lymphatic flow. CONCLUSIONS: Peripheral lymphatic function is controlled by the autonomic nervous system. In reflex sympathetic dystrophy, peripheral edema may be caused by an increased sympathetic stimulus to the lymphatics. Further study of this phenomenon may show that nuclear medicine studies, such as bone scintigraphy and lymphoscintigraphy, can be used to distinguish patients who will benefit from sympathectomy from those who will not, thereby obviating invasive testing and unnecessary invasive treatment.
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3/158. Juvenile rheumatoid arthritis and lymphoedema: lymphangiographic aspects.

    We report a 5 1/2-year-old boy with juvenile rheumatoid arthritis (JRA) and lower-limb lymphoedema. US, MRI and lymphangiography were performed. Based on the lymphangiographic study, we propose a pathogenesis based on obstruction of normal superficial lymphatic vessels in the affected limb. This is discussed with other pathogenetic factors proposed in the 16 previously reported cases of lymphoedema complicating JRA.
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4/158. Lymphoscintigraphic manifestations of Hennekam syndrome--a case report.

    Hennekam syndrome is a rare, recently described genetic disorder in which facial anomalies and mental retardation accompany congenital lymphedema and intestinal lymphangiectasia. Several other somatic abnormalities have variously been described, as have milder degrees of lymphatic dysfunction. The authors herein describe a case of Hennekam syndrome in which the diagnostic difficulties were partially overcome by the judicious use of radionuclide scintigraphy to verify the lymphedematous component of the patient's presentation.
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5/158. Multiple cutaneous malignancies arising in limbs with signs of lymphatic insufficiency in transplant patients.

    We report the cases of four renal transplant patients who developed multiple skin cancers located preferentially on lymphoedematous lower limbs. All skin tumours appeared post-transplant and the diagnosis was confirmed on histologic examination. In addition, one of the patients also underwent mastectomy with axillary dissection and radiotherapy for a left breast ductal carcinoma and subsequently developed multiple squamous carcinomas and intraepithelial carcinomas on the left upper limb.The aetiology of lymphoedema in these patients is multifactorial. In addition to immunosuppression, ultraviolet exposure, genetic factors and possibly infection with the human papilloma virus, which are thought to have a role in the aetiology of certain skin tumours, lymphoedema may have contributed to the predominant location of these skin malignancies on the lymphoedematous limbs.
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6/158. time- and dose-limiting erysipeloid rash confined to areas of lymphedema following treatment with gemcitabine--a report of three cases.

    Gemcitabine is a deoxycytidine analog with broad antitumor activity. Its main toxicities include myelosuppression, flu-like symptoms, bronchospasms and mild skin rash. We report three cases, in which the patients developed time- and dose-limiting erysipeloid skin reactions confined to areas of impaired lymphatic drainage after application of gemcitabine. Three patients with metastatic tumors (breast cancer, endometrial cancer and non-small cell lung cancer) received weekly infusions of gemcitabine (1000 mg/m2). All patients suffered from lymphedema of different origin and developed an erysipeloid erythema 40-48 h after chemotherapy within their preexisting lymphedema. Genuine erysipela was ruled out by laboratory tests and clinical observation. The skin reaction was repeatedly observed and faded after 14 days without specific treatment. Although the pathogenesis of the observed reaction is unclear, it is suspected that the skin symptoms were caused by gemcitabine or its metabolites. Gemcitabine is usually metabolized fast and excreted renally. In areas with impaired lymphatic drainage pharmakocinetics might be altered: inactivation happens slower and the drug might accumulate in the s.c. and cutaneous tissue, thus increasing local toxicity. Clinical judgement and biochemical parameters can help to tell apart genuine erysipela and the erysipeloid reaction.
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7/158. yellow nail syndrome in a 10-year-old girl.

    A 10-year-old girl with yellow dystrophic nails, bronchiectasis, chronic sinusitis and lower-limb lymphedema is presented. The underlying mechanism remains unknown although it has been postulated to be associated with lymphatic abnormalities. To date no causative treatment exists. Our patient was treated with conservative management, including a low-fat diet supplemented with medium-chain triglycerides. Moderate improvement in the lymphedema of the lower extremities was observed. To our knowledge this is the first case of yellow nail syndrome to be treated with diet.
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8/158. Successful mapping of lymphorrhea using patent blue dye after lymph node dissection for malignant melanoma.

    Patent blue is a dye that has been used for intraoperative lymphatic mapping. We used this mapping method on a patient with lymphorrhea after groin dissection. We easily detected the lymphatic channel causing lymphorrhea and successfully ligated it. This technique may have great merit for treating of lymphorrhea.
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9/158. Supermicrosurgical lymphaticovenular anastomosis for the treatment of lymphedema in the upper extremities.

    Over the last eight years, the authors analyzed obstructive lymphedema of a unilateral upper extremity in a total of 27 females, comparing the use of supramicrosurgical lymphaticovenule anastomoses and/or conservative treatment. The most common cause of edema was mastectomy, with or without subsequent radiation therapy for breast cancer. As an objective assessment of the extent of edema, the circumferences of the affected and opposite normal forearms were measured at 10 cm below the olecranon of the arm. Twelve of these patients received continual bandaging. In these patients, the average excess circumference of the affected arm was 6.4 cm over that of the normal forearm; the average duration of edema before treatment was 3.5 years; the average period for conservative treatment was 10.6 months; and the average decrease in circumference was 0.8 cm (11.7 percent of the preoperative excess). Twelve patients underwent surgery and postoperative continual bandaging. In these patients, the average excess circumference was 8.9 cm; the average duration of edema before surgery was 8.2 years; the average follow-up after surgery was 2.2 years; and the average decrease in circumference was 4.1 cm (47.3 percent of the preoperative excess). These results indicated that supermicrolymphaticovenular anastomoses with postoperative bandaging have a valuable place in the treatment of obstructive lymphedema.
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10/158. Massive localized lymphedema: additional locations and association with hypothyroidism.

    We report the second series of a new entity called "massive localized lymphedema in morbidly obese patients" (MLL), recently described in medical literature. Our 6 cases present additional locations as well as an association with hypothyroidism. Huge masses, of longstanding duration ranging from 9 months to 8 years, afflicted the thigh, popliteal fossa, scrotum, suprapubic and inguinal region, and abdomen of morbidly obese adults. Although clinical impressions were generally of a benign process, including lipoma and recurrent cellulitis, the possibility of a malignant neoplasm could not be eliminated. Poorly defined and non-encapsulated, these skin and subcutaneous lesions were most remarkable for their sheer size, measuring 50.6 cm in mean diameter (range, 38-75 cm) and weighing a mean of 6764.5 g (range, 2,060-12,000 g) The overlying skin exhibited the induration and peau d'orange characteristic of chronic lymphedema. Grossly and histologically, a prominent marbled appearance, rendered by fibrous bands intersecting lobules of adipose tissue, simulated sclerosing well differentiated liposarcoma. However, the absence of atypical stromal cells, atypical adipocytes, and lipoblasts precluded the diagnosis of well differentiated liposarcoma. Instead, reactive features, encompassing lymphatic vascular ectasia, mononuclear cell infiltrates, fibrosis, and edema between the collagen fibers, as well as ischemic changes including infarction and fat necrosis, established the diagnosis of MLL. Although the pathogenesis of MLL may be as simple as obstruction of efferent lymphatic flow by a massive abdominal pannus and/or prior surgery, the presence of hypothyroidism in 2 of our patients suggests an alternative pathogenesis. Recognition of this entity by both clinicians and pathologists should avert a misdiagnosis as a low-grade liposarcoma.
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