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Xantogranulomatosis orbitaria: Enfermedad de Erdheim-Chester
Rozas Reyes,P; Se?aris González,A; González Rodríguez,CM;
Archivos de la Sociedad Espa?ola de Oftalmología , 2004, DOI: 10.4321/S0365-66912004001000009
Abstract: case report: a patient was studied because of upper lid bilateral edema and xanthelasmae-like lesions after three years of evolution. during the ophthalmologic examination orange-yellowish lesions and two symmetrical tumours were observed on the temporal part of both upper lids. corticoid-therapy was undertaken which reduced the size of the tumours, however the size increased again after the discontinuation of treatment. a biopsy was performed and lid xanthogranulomatosis was diagnosed. other systemic examinations were normal. discussion: erdheim-chester disease is a xanthogranulomatosis that can affect ocular and periorbital structures. combination of xanthelasmae-like lesions and bilateral orbital masses should make us consider this process and try to locate any associated systemic conditions.
Xantogranulomatosis orbitaria: Enfermedad de Erdheim-Chester Orbit Xanthogranulomatosis: Erdheim-Chester disease  [cached]
P Rozas Reyes,A Se?aris González,CM González Rodríguez
Archivos de la Sociedad Espa?ola de Oftalmología , 2004,
Abstract: Caso clínico: Paciente con edema bilateral de párpados superiores y lesiones similares a xantelasmas de tres a os de evolución. La exploración oftalmológica mostraba dos tumoraciones temporales simétricas en ambos párpados superiores con lesiones naranjo-amarillentas. Se pautaron corticoides que hicieron disminuir el tama o de las tumoraciones, con recidiva de los mismos al suspender dicha medicación. La biopsia reveló una xantogranulomatosis palpebral. El resto de las exploraciones sistémicas fueron normales. Discusión: La enfermedad de Erdheim-Chester es una xantogranulomatosis que puede afectar a las estructuras oculares y periorbitarias. La combinación de lesiones similares a xantelasmas y masas orbitarias bilaterales deben hacernos pensar en este proceso y buscar la posible afectación sistémica. Case report: A patient was studied because of upper lid bilateral edema and xanthelasmae-like lesions after three years of evolution. During the ophthalmologic examination orange-yellowish lesions and two symmetrical tumours were observed on the temporal part of both upper lids. Corticoid-therapy was undertaken which reduced the size of the tumours, however the size increased again after the discontinuation of treatment. A biopsy was performed and lid xanthogranulomatosis was diagnosed. Other systemic examinations were normal. Discussion: Erdheim-Chester disease is a xanthogranulomatosis that can affect ocular and periorbital structures. Combination of xanthelasmae-like lesions and bilateral orbital masses should make us consider this process and try to locate any associated systemic conditions.
Unusual manifestation of Erdheim-Chester disease
Antony Pan, Terence Doyle, Martin Schlup, Ralf Lubcke, Michael Schultz
BMC Gastroenterology , 2011, DOI: 10.1186/1471-230x-11-77
Abstract: This case report describes the case of a 69-year-old man who presented otherwise well to the gastroenterology department with unspecific abdominal symptoms, nausea, vomiting and weight loss. ECD involving the gastrointestinal tract was confirmed clinically, radiologically and histologically.Gastroenterological manifestation of ECD is rare but should be considered in the differential diagnosis in patients presenting with evidence of multi-organ disease and typical radiological features of Erdheim-Chester disease elsewhere.Erdheim-Chester Disease (ECD) is a rare multisystem histiocytosis characterized by the xanthomatous or xanthogranulomatous infiltration of tissues with histiocytes, surrounded by fibrosis. The disease can affect multiple organs systems, but gastrointestinal involvement, is exceedingly rare. We describe here the case of a 69-year old man with ECD who presented to the gastroenterology department with unspecific abdominal symptoms, nausea, vomiting and weight loss.A previously fit and well 69-year-old man was admitted to the gastroenterology department with a short one month history of lethargy, decreased appetite, persistent vomiting, significant weight loss of six kilograms over 1 month and a dry cough. He denied abdominal pain, haematemesis, dysphagia and a change in bowel habit and described no cardiac, respiratory, neurological symptoms or bone pain. His past medical history includes appendectomy and total hip joint replacement. At the time of admission, he was not on any regular medication.Physical examination revealed that he was febrile (38.8C°) and appeared cachexic but with no peripheral stigmata of chronic liver disease. His abdominal examination revealed hepatomegaly but no other organomegaly, per rectal examination was unremarkable. His cardiopulmonary examinations were unremarkable apart from bilateral pitting edema up to his ankles. Peripheral blood analysis revealed an anaemia of chronic disease with hemoglobin of 108 g/L (norm - male 1
Erdheim-Chester Disease Associated with Marginal Zone Lymphoma and Monoclonal Proteinemia
Peter G. Pavlidakey,Alok Mohanty,Lisa J. Kohler,Howard J. Meyerson
Case Reports in Hematology , 2011, DOI: 10.1155/2011/941637
Abstract: Erdheim-Chester disease (ECD) is a rare non-Langerhans cell histiocytosis. We report a fatal case of ECD with extensive cardiac involvement associated with a marginal zone lymphoma and monoclonal proteinemia in a young man. This is the first reported association of ECD with a monoclonal gammopathy or a lymphoma.
Doen?a de Erdheim-Chester: relato de dois casos
Hexsel, Fernando Fernandez;Canazaro, Letícia Frigo;Capoani, Mariana;Aguiar, Paula Musa;Suwa, Eiji;Maciel, Antonio Carlos;
Radiologia Brasileira , 2009, DOI: 10.1590/S0100-39842009000400015
Abstract: erdheim-chester disease is a rare non-langerhans cell histiocytosis of unknown etiology, affecting multiple organ system, involving bones, central nervous system, eyes, lungs, mediastinum, kidneys and retroperitoneum. the authors report two cases that progressed with the typical presentation of the disease. radiological findings were in agreement with literature and guided the diagnosis, confirmed by immunohistochemistry.
Bilateral orbital involvement in Erdheim-Chester disease: case report
Monteiro, Mário Luiz Ribeiro;Sampaio, César Moreira;
Arquivos Brasileiros de Oftalmologia , 2002, DOI: 10.1590/S0004-27492002000600015
Abstract: purpose: to report a case of erdheim-chester disease with bilateral orbital involvement and to inform ophthalmologists about this uncommon condition. methods: case report. a 41-year-old man presented with proptosis, lid xanthelasmas, disc edema and extraocular muscle restrictions. he was suspected of having a systemic lymphoma and had already been submitted to a retroperitoneal biopsy, however, the diagnosis was still uncertain. results: the patient was evaluated by computed tomography and magnetic resonance imaging of the orbit. orbital pseudotumor was suspected and a biopsy of the orbital lesion initially failed to provide the correct diagnosis which was only made after detailed analysis of the clinical and imaging findings, and review of the histopathologic studies. the systemic diagnosis was made after the orbital diagnosis although the patient had been extensively investigated for more than one year. conclusions: erdheim-chester disease is a rare idiopathic systemic condition characterized by a xanthogranulomatous process involving retroperitoneum, heart, lungs, bone and other tissues. the condition is often fatal due to renal or cardiovascular complications. ocular findings are rare but may be very helpful for the diagnosis. therefore, ophthalmologists should be aware of the clinical manifestations and imaging findings of this rare disease in order to establish an early diagnosis.
Bilateral orbital involvement in Erdheim-Chester disease: case report  [cached]
Monteiro Mário Luiz Ribeiro,Sampaio César Moreira
Arquivos Brasileiros de Oftalmologia , 2002,
Abstract: Purpose: To report a case of Erdheim-Chester disease with bilateral orbital involvement and to inform ophthalmologists about this uncommon condition. Methods: Case report. A 41-year-old man presented with proptosis, lid xanthelasmas, disc edema and extraocular muscle restrictions. He was suspected of having a systemic lymphoma and had already been submitted to a retroperitoneal biopsy, however, the diagnosis was still uncertain. Results: The patient was evaluated by computed tomography and magnetic resonance imaging of the orbit. Orbital pseudotumor was suspected and a biopsy of the orbital lesion initially failed to provide the correct diagnosis which was only made after detailed analysis of the clinical and imaging findings, and review of the histopathologic studies. The systemic diagnosis was made after the orbital diagnosis although the patient had been extensively investigated for more than one year. Conclusions: Erdheim-Chester disease is a rare idiopathic systemic condition characterized by a xanthogranulomatous process involving retroperitoneum, heart, lungs, bone and other tissues. The condition is often fatal due to renal or cardiovascular complications. Ocular findings are rare but may be very helpful for the diagnosis. Therefore, ophthalmologists should be aware of the clinical manifestations and imaging findings of this rare disease in order to establish an early diagnosis.
Fever of unknown origin as the first manifestation of Erdheim-Chester disease  [PDF]
Ning Li, Mingquan Chen, Huaping Sun, Yun Bao, Jiming Zhang
Case Reports in Clinical Medicine (CRCM) , 2013, DOI: 10.4236/crcm.2013.26095
Abstract: Erdheim-Chester disease (ECD) is an uncommon non-Langerhans cell histiocytosis that affects multiple body systems and can present clinically in a myriad of ways. We report the case of a 29-year-old man presented with fever and diffuse bone pain. Physical examination showed bilateral and symmetrical long bone pain, especially the knees. Radiographs showed multiple mixed bone lesions with sclerotic and lytic areas of the femora, tibiae, and fibula. Biopsy of the tibial area displayed foamy lipid-laden histiocytes, confirming the diagnosis of ECD.
Enfermedad de Erdheim-Chester e histiocitosis de células de Langerhans: ?Una asociación fortuita?
Simiele Narvarte,A.; Novoa Sanjurjo,F.; Gómez Rodríguez,N.; Antón Badiola,I.;
Anales de Medicina Interna , 2004, DOI: 10.4321/S0212-71992004001200006
Abstract: erdheim-chester disease is an idiopathic systemic histiocytosis hystologically different to langerhans histiocytosis which is characterized by symmetric sclerotic bone lesions, predominantly affecting the diaphyseal and metaphyseal areas of large bones. it is not classified within malignant histicytosis, but the clinical course tends to be agressive with a poor reponse to different schedules of treatment. in rare cases the association of both entities has been reported: erdheim-chester disease and langerhans histiocytosis. we present a new case with histological data of both histiocytosis whose clinical course included bone and muscle pain, insipidus diabetes, exophthalmos, bilateral symmetrical sclerosing bone lesions and a cerebellar syndrome.
Acometimento intra-ocular na doen?a de Erdheim-Chester - primeiro relato na literatura: relato de caso
Biccas Neto, Laurentino;Zanetti, Fernando;
Arquivos Brasileiros de Oftalmologia , 2007, DOI: 10.1590/S0004-27492007000500025
Abstract: erdheim-chester disease (ecd) is a granulomatous and infiltrative disorder of unknown etiology with proliferation of cholesterol-containing histiocytes and peculiar bone involvement. it is very similar to langerhans cell histiocytosis (lch) on histology but with a different immunohistochemical profile. this is the first report of intraocular involvement in this disease. mpsg, a 46 y.o. woman, presented with proptosis of the od. she referred ulcerated lesions on the hard palate, symmetrical and bilateral osteosclerosis of the fibulae and tibiae and a nodule in the right breast (biopsy: xantomatous histiocytic infiltrate cd68+, s-100 and cd1a negative on immunohistochemistry compatible with ecd). mri studies demonstrated an extraconal tumor in the juxta-bulbar temporal portion of the right orbit close to the lacrimal gland and hyperintense on t1. vision was 20/20 ou, with numerous drusen in the posterior pole, similar to basal laminar drusen. two regions of orange subretinal infiltrates that showed progressive staining on the angiogram were seen in the peripapillary region and also close to the fovea in the od. choroidal neovascular membranes were seen 2 years later in ou leading to severe visual loss in the os and to a slight visual field loss in the od, which retained 20/20 vision. this pioneer report depicts in vivo characteristics of histiocytic granulomas in ecd. caution should be taken with patients with ecd as potentially blinding intraocular complications may arise.
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