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Surgical management of persistent, symptomatic nonvenereal sclerosing lymphangitis of the penis.

Nonvenereal sclerosing lymphangitis of the penis is a rare condition involving the distal lymphatics of the penis. Its association with trauma has been shown. Although there is a minimal inflammatory response the etiology is unknown. Clinically, the patient notices a nontender, serpiginous nodular mass just proximal to the coronal sulcus. Most cases are self-limited and conservative management is indicated. Surgical excision is warranted for persistently symptomatic lesions.

Adult↗

[Nocardia brasiliensis leg ulcer and nodular lymphangitis in France].

INTRODUCTION: Nocardia brasiliensis is a very rarely reported cause of chronic phagedenic ulcerations. We report the case of an elderly woman who developed such an infection after falling on her right leg on the road in the Bresse country (an essentially agricultural and bovine-cattle breading region) and developed a chronic phagedenic ulcer secondarily complicated by nodular lymphangitis of the thigh. CASE REPORT: A 75 year-old woman fell on her right leg on the side of the main road outside her hamlet in the Bresse country and secondarily developed a chronique phagedenic ulceration. We first considered her as suffering from pyoderma gangrenosum. A complete scanning only revealed an autoimmune thyroiditis and a rapidly healing gastric ulceration, and none of the treatments, either local or systemic, helped the skin condition to heal. After 3 weeks of application of a local corticoid ointment, the patient developed fever, general malaise, an exacerbation of her wound and an infiltration of the skin round her knee, together with nodular lymphangitic dissemination. A supplementary bacterial swab disclosed massive proliferation of a slow-growing Gram-positive bacillus, which proved to be Nocardia brasiliensis, together with a methicillino-sensitive Staphylococcus aureus. The treatment with sulfamethoxazole-trimetoprim gave a rash after 12 hours and was changed to amoxicillin and clavulanic acid, which rapidly proved to be permanently effective. DISCUSSION: The revelation of this particular slow-growing bacteria is difficult and requires bacterial swabs. Nocardia brasiliensis is relatively rare in primary skin ulcerations and we discuss the reasons why an elderly women should find this bacteria on the road outside her hamlet in the French countryside. This particular infectious condition requires general scanning, to make sure that the primary skin condition does not extend to other organs. We review the therapeutical options for patients who exhibit allergic reactions to the classically effective antibiotic drugs.

Accidental Falls↗

Sclerosing lymphangitis of the penis.

Nonvenereal sclerosing lymphangitis is a rare penile lesion consisting of a minimally tender, indurated cord involving the coronal sulcus and occasionally adjacent distal penile skin. This disorder most often occurs after vigorous sexual activity and resolves spontaneously. However, as evidenced by 2 of the 3 cases reported, this condition also may be associated with underlying sexually transmitted disease that necessitates specific therapy.

Adult↗

[Non-venereal sclerosing lymphangitis of the penis. Report of two clinical cases].

Nonvenereal sclerosing lymphangitis of the penis is a rare condition that preferably affects men aged between 20 and 40 years, and it is appeared like a hard cord or a nodular lesion in the sulcus coronarius penis. It is believed that it has origin in lymph vessels of the penis. Usually it is resolved in a self-limited way, so the initial treatment is conservative. We report two cases, and some etiopathogenic, diagnosis and therapeutic aspects are discussed.

Adult↗

Cutaneous histiocytic lymphangitis: an unusual manifestation of rheumatoid arthritis.

Two cases are presented of unusual cutaneous lesions associated with rheumatoid arthritis in underlying joints. The lesions were evanescent, erythematous and violaceous partly macular and partly indurated plaques, with a livedo-like pattern of erythema at the edge in one case. Histological changes were identical in the two cases. The major features were dilated, dermal lymphatics containing aggregates of inflammatory cells, mainly histiocytes, with adjacent perivascular lymphoid aggregates. An appropriate name for this reaction would appear to be cutaneous histiocytic lymphangitis.

Arthritis, Rheumatoid↗

Lymphangitis carcinomatosis of unknown origin presenting as severe pulmonary hypertension.

An apparently healthy 46-year-old woman was admitted because of progressive shortness of breath that had begun 2 months before her admission. Physical examination revealed a patient with respiratory distress, tachycardia, and mild jugular venous distention; otherwise, results were unremarkable. Our investigation revealed hypoxia and severe pulmonary hypertension with signs of right heart dysfunction, but no primary cause was found. The patient died 5 days after admission. Autopsy revealed pulmonary lymphangitis carcinomatosis caused by papillary carcinoma. No primary tumor was found.

Carcinoma, Papillary↗

Sclerosing lymphangitis of the penis: a lymphangiofibrosis thrombotica occlusiva.

Three patients with so-called non-venereal sclerosing lymphangitis of the penis are presented. Light and electron microscopy of one case revealed a lymphatic collecting vessel with a fibrin thrombus in the process of recanalization and vessel wall fibrosis due to hyperplasia of smooth muscle cells and fibroblasts. The term 'lymphangiofibrosis thrombotica occlusiva' is proposed. Lymph stasis is suggested as a provoking factor for the dilatation and clinically striking firm thickening of the affected collecting vessel. No microorganisms were recognized. Within the fibrin thrombus, sprouts of endothelial cells showed intracellular vacuoles, probably indicating the first identifiable step in lymph capillary lumen formation. Signs of collagen remodelling were encountered in the thickened vessel wall.

Adult↗

Pulmonary lymphangitis carcinomatosa and acute pancreatitis: a rare presentation of choledochal cyst.

Pulmonary lymphangitis carcinomatosa is an unusual cause of death in a young adult. This case describes an apparently healthy young woman who presented with severe acute pancreatitis, which is a recognized complication of a choledochal cyst. Autopsy examination revealed advanced malignancy with poorly differentiated adenocarcinoma penetrating the wall of the choledochal cyst and metastatic adenocarcinoma in the lymph nodes, lungs and kidneys. This case emphasises the unusual presentation of a choledochal cyst with acute pancreatitis and the aggressive nature of malignancy associated with this congenital anomaly.

Acute Disease↗

A unique case of eccrine porocarcinoma with pulmonary lymphangitis and pericardial involvement: biological characterization and clinical aggressiveness.

A unique case of eccrine porocarcinoma with pulmonary lymphangitis and pericardial involvement is reported. The clinical course was aggressive, leading to the death of the patient a few months after diagnosis. Certain pathologial markers of clinical aggressiveness were retrospectively investigated: p53 and Ki-67 expression were determined by means of immunohistochemistry. Angiogenesis was assessed by determination of intratumor microvessel density at the vascular 'hot spot' with the anti-CD34 monoclonal antibody and quantitative analysis using computerized image analyzer. Both primary tumor and metastatic lymph node presented immunostaining for p53 and Ki-67, with a higher degree of vascularization in the secondary lesions compared to the primary tumor. Our findings suggest a correlation between tumor vascularization and clinicopathological parameters of aggressiveness in malignant eccrine porocarcinoma. Taking into account the disappointing results of current treatments for metastatic eccrine porocarcinoma, the assay of microvessel density may be helpful in selecting the patients of high risk for recurrence or death who may benefit of anti-angiogenic therapies.

Acrospiroma↗

Cutaneous metastatic lymphangitis from squamous cell carcinoma of the cervix.

A case of cutaneous metastatic lymphangitis which appeared during the unusual evolution of a cervix carcinoma is reported. DNA sequences of No. 18 human papilloma virus (HPV) were isolated in both vaginal and cutaneous specimens. The hypothesis that a particular evolution of invasive cervix carcinomas might be related to the type of associated HPV is suggested.

Adult↗

Lipogranulomatous lymphangitis in canine intestinal lymphangiectasia.

Lipogranulomatous lymphangitis of the intestine occurred in four dogs with intestinal lymphangiectasia. All four presented with chronic diarrhea; three had ascites and two had hypoalbuminemia. Lipogranulomas appeared in lymphatics, often at the point of mesenteric attachment, of small intestine, ileum, or ileum and colon. Mesenteric lymphatics were obstructed and villous lacteals were distended. Mesenteric lymph nodes of one dog contained large lipid spaces and that same animal had a solitary subcapsular lipogranuloma of the liver. This disorder is one of several that result in protein-losing enteropathy in dogs.

Animals↗

Carcinomatous lymphangitis mimicking pulmonary thromboembolism.

A 41-year-old woman was admitted with rapidly worsening dyspnea. Echocardiography disclosed interventricular septal flattening and a markedly decreased left ventricle, although left ventricular contraction remained normal. Computed tomography of the chest demonstrated slightly dilated main pulmonary arteries and fine reticulonodular densities in the lung. Examination of a transbronchial lung biopsy specimen revealed carcinomatous lymphangitis, and the patient died 7 days after admission. The clinical presentation of this patient was difficult to discriminate from that seen with pulmonary thromboembolism.

Adenocarcinoma↗

Sudden onset of thrombocytopenia after the treatment with chemotherapeutic agent(s) in a patient with lymphangitis carcinomatosa.

Cisplatin (CDDP 100 mg/m2, div), adriamycin (ADR 20 mg/body, iv) and futrafur-uracil conjugate (UFT 200 mg/day, po) were administered on the same day to a 51-year-old housewife with lymphangitis carcinomatosa of the lung. During the CDDP infusion, the patient unexpectedly became highly febrile. On the following day, the patient developed purpura on the upper and lower extremities, and her platelet count was 3.2 x 10(4)/mm3 in spite of being 32.4 x 10(4)/mm3 on the day before chemotherapy. The platelet count recovered to 9.4 x 10(4)/mm3 on day 3, and increased to a maximum of 56.1 x 10(4)/mm3 on day 20. Previously, we had treated several patients with adenocarcinoma by the same protocol, but never experienced such a case that developed a sudden fall in their platelet count. Although the true mechanisms remain to be solved, this case is very interesting from a standpoint of drug-induced thrombocytopenia.

Adenocarcinoma↗

Lymphangitis carcinomatosa.

A case of lymphangitis carcinomatosa secondary to a primary gastric adenocarcinoma is reported. Diagnosis was suspected clinically and radiologically, but was established only at autopsy. The condition is reviewed. When suspected, transbronchial lung biopsy would allow histological confirmation of the diagnosis.

Adenocarcinoma↗

[Nonvenereal lymphangitis of the penis].

Two personal cases of Non venereal Lymphangitis of the Penis are presented in 23 and 28 men, and 38 cases from the literature are reviewed. This benign self-limiting condition is not too well known among dermatologists and urologists. The clinical changes, in particular noted by palpation are characteristic: painless firm cartilaginous cord freely movable behind the corona of glans penis in the coronary sulcus. The inguinal lymph nodes are not involved. The microscopic features in one of our patients demonstrated that the lesion results from cystic dilatation of secondary organization of the thrombus. The etiology is unknown. We favour the mecanical trauma as an important factor based on excessive sexual activity of the patients.

Adult↗

[Carcinomatous lymphangitis].

DEFINITION: Carcinomatous lymphangitis is a radioclinical entity accounting for about 8% of all cases of lung metastasis defined as the presence of tumoral cells in lymph vessels and lung interstitium. DIAGNOSIS: Biopsy specimens or bronchial brushings obtained by fibroendoscopy or bronchioalveolar lavage fluid usually reveal adenocarcinoma. PRACTICAL MANAGEMENT: In clinical practice, the patient presents with dyspnea and non-specific infiltration on the chest x-ray. The clinical situation worsens rapidly. Millimetric CT-scan shows highly suggestive polygonal images in the subpleural area. Respiratory function tests may be helpful for the differential diagnosis, particularly in difficult cases, showing a mixed ventilation disorder without altered carbon monoxide diffusion and hypoxemia at rest without hypercapnia. SEARCH FOR THE PRIMARY CANCER: Primary lesions must be identified for specific treatment. Pathology findings help guide the search. Despite the highly unfavorable prognosis (median survival = 3 months), etiological treatment when possible can improve quality of life and possibly survival. Symptomatic treatment is indicated and must be adapted to each individual case.

Adenocarcinoma↗