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[PMID]:29048579
[Au] Autor:Tsukamoto S; Righi A; Vanel D; Honoki K; Donati DM; Errani C
[Ad] Dirección:Department of Orthopaedic Surgery, Nara Medical University, Kashihara, Nara, Japan.
[Ti] Título:Development of high-grade osteosarcoma in a patient with recurrent giant cell tumor of the ischium while receiving treatment with denosumab.
[So] Fuente:Jpn J Clin Oncol;47(11):1090-1096, 2017 Nov 01.
[Is] ISSN:1465-3621
[Cp] País de publicación:England
[La] Idioma:eng
[Ab] Resumen:Malignant transformation of giant cell tumor of bone (GCTB) without radiotherapy exposure is exceptionally rare, occurring in less than 1% of GCTBs. The safety and efficacy of denosumab in patients with GCTB was recently reported. We herein report a case of a benign recurrent GCTB with an H3F3A mutation that underwent secondary malignant transformation during treatment with denosumab. A 29-year-old woman underwent curettage of a GCTB of the left ischium in 2005. Ten years after the first surgery, the GCTB recurred locally. We started treatment with denosumab. During the first 5 months of treatment, we observed a demarcated area of osteosclerosis in the recurrent lesion, and the patient's clinical condition improved. At 6 months, however, the patient developed pain, and a rapidly growing mass was detected by computed tomography. An incisional biopsy was performed. Histologic analysis showed a high-grade osteosarcoma. The patient developed lung metastases and died soon after beginning chemotherapy. The mechanism of sarcomatous transformation of GCTB during denosumab therapy is unclear. These findings suggest that the scientific community should be aware of the possible malignant transformation of GCTB during denosumab treatment.
[Pt] Tipo de publicación:JOURNAL ARTICLE
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171019
[Lr] Fecha última revisión:171019
[St] Status:In-Process
[do] DOI:10.1093/jjco/hyx112


  2 / 4634 MEDLINE  
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[PMID]:29019884
[Au] Autor:Hara D; Akiyama H; Nukui S; Shimizu T; Hoshikawa M; Hasegawa Y
[Ad] Dirección:aDepartment of Internal Medicine, Division of Neurology bDepartment of Diagnostic Pathology, Division of Diagnostic Pathology, St. Marianna University School of Medicine, Kawasaki, Kanagawa, Japan.
[Ti] Título:Utility of osteosclerotic lesion biopsy in diagnosis of POEMS syndrome: A case report.
[So] Fuente:Medicine (Baltimore);96(41):e8188, 2017 Oct.
[Is] ISSN:1536-5964
[Cp] País de publicación:United States
[La] Idioma:eng
[Ab] Resumen:RATIONALE: We report a case of successful diagnosis of POEMS (polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes) syndrome based on monoclonality that was confirmed by an osteosclerotic lesion biopsy in a patient without pathognomonic symptoms or monoclonal gammopathy, probably because of comorbidities, which included systemic lupus erythematosus, rheumatoid arthritis, and Sjögren syndrome. PATIENT CONCERNS: A 57-year-old woman presented with an approximately 2-year history of numbness in the toes that had gradually spread, along with muscle weakness in both arms and legs. She had been receiving immunosuppressant and corticosteroid therapy since being diagnosed with systemic lupus erythematosus and Sjögren syndrome at the age of 31 years and rheumatoid arthritis at the age of 44 years. Neurological examination revealed predominantly distal hypoesthesia and weakness in a typical stocking-and-glove pattern. Immunoelectrophoresis revealed elevated polyclonal immunoglobulin, which was attributed to her known underlying disease. DIAGNOSES: Biopsy of an osteosclerotic lesion confirmed proliferation of monoclonal plasma cells, leading to a diagnosis of POEMS syndrome. INTERVENTIONS AND OUTCOMES: Lenalidomide therapy was started after the diagnosis and the patient had a favorable outcome. LESSONS: Osteosclerotic lesion biopsy can be useful for diagnosis of POEMS syndrome in difficult cases.
[Mh] Términos MeSH primario: Osteosclerosis
Síndrome POEMS
Células Plasmáticas
Talidomida/análogos & derivados
[Mh] Términos MeSH secundario: Artritis Reumatoide/complicaciones
Artritis Reumatoide/quimioterapia
Proliferación de la Célula
Diagnóstico Diferencial
Femenino
Humanos
Inmunoelectroforesis/métodos
Factores Inmunológicos/administración & dosificación
Lupus Eritematoso Sistémico/complicaciones
Lupus Eritematoso Sistémico/quimioterapia
Mediana Edad
Examen Neurológico/métodos
Osteosclerosis/diagnóstico por imagen
Osteosclerosis/patología
Síndrome POEMS/diagnóstico
Síndrome POEMS/quimioterapia
Síndrome POEMS/fisiopatología
Células Plasmáticas/inmunología
Células Plasmáticas/patología
Síndrome de Sjögren/complicaciones
Síndrome de Sjögren/quimioterapia
Talidomida/administración & dosificación
Resultado del Tratamiento
[Pt] Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE
[Nm] Nombre de substancia:
0 (Immunologic Factors); 4Z8R6ORS6L (Thalidomide); F0P408N6V4 (lenalidomide)
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171019
[Lr] Fecha última revisión:171019
[Sb] Subgrupo de revista:AIM; IM
[Da] Fecha de ingreso para procesamiento:171011
[St] Status:MEDLINE
[do] DOI:10.1097/MD.0000000000008188


  3 / 4634 MEDLINE  
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[PMID]:28291506
[Au] Autor:Philipponnet C; Kemeny JL; Garrouste C; Soubrier M; Heng AE
[Ti] Título:Immunotactoid glomerulopathy leading to the discovery of POEMS syndrome
.
[So] Fuente:Clin Nephrol;87(6):310-315, 2017 Jun.
[Is] ISSN:0301-0430
[Cp] País de publicación:Germany
[La] Idioma:eng
[Ab] Resumen:Monoclonal gammopathy of renal significance (MGRS) can manifest in many different ways depending on the nature of the immunoglobulin and its physicochemical properties. MGRS can lead to the discovery of a hematological malignancy. We report the case of a 32-year-old female patient who underwent renal biopsy on account of an impure nephrotic syndrome associated with immunoglobulin (Ig)G κ monoclonal gammopathy. Histological analysis revealed membranoproliferative glomerulonephritis with IgG, IgM, κ, λ, and C3 deposits. Due to an unfavorable progression, a second renal biopsy was performed. Electron microscopy analysis revealed an immunotactoid glomerulopathy. At the same time, a POEMS syndrome diagnosis (polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin abnormalities) was confirmed in light of the following: 1) IgG κ monoclonal gammopathy, 2) axonal neuropathy, 3) osteosclerosis, 4) melanoderma, 5) hepatosplenomegaly and adenopathies, 6) Castleman disease, and 7) edema. Our observation is the first case of immunotactoid glomerulopathy leading to the discovery of a POEMS syndrome. Renal involvement in POEMS syndrome typically exhibits a thrombotic microangiopathy-like membranoproliferative glomerulonephritis appearance associated with endothelial lesions stigmata. However, monoclonal immunoglobulin deposition disorder should be considered in the event of an atypical case. In this indication, electron microscopy is the examination of choice for assessing immunoglobulin deposition nephropathy.
.
[Mh] Términos MeSH primario: Glomerulonefritis Membranoproliferativa
Síndrome POEMS
Paraproteinemias
[Mh] Términos MeSH secundario: Adulto
Femenino
Humanos
[Pt] Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171018
[Lr] Fecha última revisión:171018
[Sb] Subgrupo de revista:IM
[Da] Fecha de ingreso para procesamiento:170314
[St] Status:MEDLINE
[do] DOI:10.5414/CN109056


  4 / 4634 MEDLINE  
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[PMID]:28725909
[Au] Autor:Peicher K; Maalouf NM
[Ad] Dirección:Division of Endocrinology, Department of Internal Medicine, University of Texas Southwestern Medical Center, Dallas, TX, USA.
[Ti] Título:Skeletal Fluorosis Due to Fluorocarbon Inhalation from an Air Dust Cleaner.
[So] Fuente:Calcif Tissue Int;101(5):545-548, 2017 Nov.
[Is] ISSN:1432-0827
[Cp] País de publicación:United States
[La] Idioma:eng
[Ab] Resumen:Skeletal fluorosis (SF) is an osteosclerotic metabolic bone disorder caused by excessive ingestion or inhalation of fluoride. SF is extremely rare in developed countries. We report a case of SF due to inhalational abuse from a fluoride-containing air dust cleaner. A 33-year-old man with no past medical history presented with progressively worsening low back pain for 2 years. Physical examination was notable for loss of lumbar lordosis and tenderness over the lumbar spine. Radiographs were notable for uniform generalized osteosclerosis in the long bones, entire spine, rib cage, and pelvic bones, and loss of the normal lumbar curvature. DXA scan showed Z-scores of +10.7 at the lumbar spine, +6.5 at the total hip, and +1.0 at the 1/3 radius. Laboratory studies were notable for elevated serum alkaline phosphatase (334 U/L, ref: 40-129 U/L) compared to a normal value 3 years prior, suggesting acquired osteosclerosis. Serum fluoride concentration returned elevated (2.8 mg/L, ref: 0.0-0.2 mg/L). Initially, the source of fluoride excess could not be identified. At a follow-up visit, he was found inhaling from a can of an air duster hidden in an inner pocket. He admitted "huffing" 2-7 cans weekly from a fluorocarbon-containing air dust cleaner for the past 3 years to achieve a euphoric feeling, explaining the source of his SF. Fluoride inhalation can be a potential source for SF, and should be suspected in patients with acquired osteosclerosis, as inhalant abuse is increasingly practiced in many countries.
[Pt] Tipo de publicación:JOURNAL ARTICLE
[Em] Mes de ingreso:1707
[Cu] Fecha actualización por clase:171016
[Lr] Fecha última revisión:171016
[St] Status:In-Process
[do] DOI:10.1007/s00223-017-0305-0


  5 / 4634 MEDLINE  
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[PMID]:28233026
[Au] Autor:Saglam D; Bilgici MC; Bekçi T; Albayrak C; Albayrak D
[Ad] Dirección:Department of Radiology, School of Medicine, Ondokuz Mayis University, Kurupelit, Samsun, Turkey. dilekuzman@hotmail.com.
[Ti] Título:Autosomal recessive osteopetrosis with a unique imaging finding: multiple encephaloceles.
[So] Fuente:Skeletal Radiol;46(5):701-704, 2017 May.
[Is] ISSN:1432-2161
[Cp] País de publicación:Germany
[La] Idioma:eng
[Ab] Resumen:Osteopetrosis is a hereditary form of sclerosing bone dysplasia with various radiological and clinical presentations. The autosomal recessive type, also known as malignant osteopetrosis, is the most severe type, with the early onset of manifestations. A 5-month-old infant was admitted to our hospital with recurrent respiratory tract infections. Chest X-ray and skeletal survey revealed the classic findings of osteopetrosis, including diffuse osteosclerosis and bone within a bone appearance. At follow-up, the patient presented with, thickened calvarium, multiple prominent encephaloceles, and dural calcifications leading to the intracranial clinical manifestations with bilateral hearing and sight loss. Autosomal recessive osteopetrosis is one of the causes of encephaloceles and this finding may become dramatic if untreated.
[Mh] Términos MeSH primario: Encefalocele/complicaciones
Encefalocele/diagnóstico por imagen
Osteopetrosis/complicaciones
Osteopetrosis/diagnóstico por imagen
[Mh] Términos MeSH secundario: Encéfalo/diagnóstico por imagen
Humanos
Lactante
Imagen por Resonancia Magnética
Costillas/diagnóstico por imagen
Columna Vertebral/diagnóstico por imagen
Tomografía Computarizada por Rayos X
[Pt] Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171012
[Lr] Fecha última revisión:171012
[Sb] Subgrupo de revista:IM
[Da] Fecha de ingreso para procesamiento:170224
[St] Status:MEDLINE
[do] DOI:10.1007/s00256-017-2595-8


  6 / 4634 MEDLINE  
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[PMID]:27966515
[Au] Autor:Naser AZ; Roshanzamir N
[Ad] Dirección:Department of Oral and Maxillofacial Radiology, Dental School, Isfahan University of Medical Sciences, Isfahan, Iran.
[Ti] Título:Prevalence of idiopathic osteosclerosis in an Iranian population.
[So] Fuente:Indian J Dent Res;27(5):544-546, 2016 Sep-Oct.
[Is] ISSN:1998-3603
[Cp] País de publicación:India
[La] Idioma:eng
[Ab] Resumen:AIM: Idiopathic osteosclerosis (IO) is a localized radiopacity with no associated pain, bony expansion, symptom, or known etiology. The aim of this study was to assess its frequency and distribution according to its location and patient's age and gender and internal structure using panoramic radiography. SUBJECTS AND METHODS: In this retrospective study, 3975 panoramic radiographs (PRs) were used. Radiopaque regions which had no certain relationship to any known sources of hard tissue production were accepted as IO and characteristics such as shape, location, internal structure, and personal information were investigated. STATISTICAL ANALYSIS USED: The obtained data were evaluated using SPSS software and Chi-square test. RESULTS: IO was found in 113 (2.84%) out of 3975 patients. The most frequent site was the posterior region of mandible. There was no significant difference in the incidence of IO between genders (P = 0.766). CONCLUSION: IO is an asymptomatic radiopaque mass commonly seen in PR, which was found mostly in the first molar region of mandible. Furthermore, its maximum frequency was in the second decade.
[Mh] Términos MeSH primario: Enfermedades Maxilomandibulares/epidemiología
Osteosclerosis/epidemiología
[Mh] Términos MeSH secundario: Adolescente
Adulto
Factores de Edad
Niño
Femenino
Humanos
Irán/epidemiología
Enfermedades Maxilomandibulares/diagnóstico por imagen
Masculino
Mediana Edad
Osteosclerosis/diagnóstico por imagen
Prevalencia
Radiografía Panorámica
Estudios Retrospectivos
Factores Sexuales
Adulto Joven
[Pt] Tipo de publicación:JOURNAL ARTICLE
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171012
[Lr] Fecha última revisión:171012
[Sb] Subgrupo de revista:D
[Da] Fecha de ingreso para procesamiento:161214
[St] Status:MEDLINE
[do] DOI:10.4103/0970-9290.195679


  7 / 4634 MEDLINE  
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[PMID]:28393275
[Au] Autor:Rathod AK; Dhake RP; Borde MD
[Ad] Dirección:Department of Orthopaedics, LTMMC & LTMGH, First floor, College building, Sion hospital, Sion west, Mumbai, Maharashtra, 400022, India.
[Ti] Título:Traumatic multiple cervical spine injuries in a patient with osteopetrosis and its management.
[So] Fuente:Eur Spine J;26(Suppl 1):229-235, 2017 May.
[Is] ISSN:1432-0932
[Cp] País de publicación:Germany
[La] Idioma:eng
[Ab] Resumen:STUDY DESIGN: Single case report. OBJECTIVE/PURPOSE: To report multiple level fractures of cervical spine in a patient with osteopetrosis and its management. Osteopetrosis is a rare inherited condition characterized by defective remodeling resulting in hard and brittle bones with diffuse osteosclerosis. Fractures of spine are rare as compared to the common long bone fractures. We report a case of traumatic multiple level fractures of cervical spine in osteopetrosis and its management which has rarely been reported in the literature before, if any. METHODS: 17-year-old boy presented with severe tenderness in neck and restricted range of motion following a trivial injury to the neck in swimming pool. The neurology was normal and he was diagnosed to have autosomal dominant osteopetrosis on evaluation. Imagining findings, clinical course and the method of treatment are discussed. RESULTS: Radiological evaluation revealed presence of multiple level fractures of cervical vertebrae with end plate sclerosis. Patient was managed with cervical skeletal traction in appropriate extension position for 6 weeks followed by hard cervical collar for another 6 weeks. Follow-up radiographs at 18 months and 2.5 years showed healed fractures with no residual instability or symptoms. CONCLUSION: The case report discusses rare occurrence of multiple level fractures of cervical spine following trivial injury to the neck in a patient with osteopetrosis and its treatment with conservative management.
[Pt] Tipo de publicación:JOURNAL ARTICLE
[Em] Mes de ingreso:1704
[Cu] Fecha actualización por clase:171011
[Lr] Fecha última revisión:171011
[St] Status:In-Process
[do] DOI:10.1007/s00586-017-5083-x


  8 / 4634 MEDLINE  
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[PMID]:28186845
[Au] Autor:Obinata K; Shirai S; Ito H; Nakamura M; Carrozzo M; Macleod I; Carr A; Yamazaki Y; Tei K
[Ad] Dirección:1 Omni Dentix, Dartmouth, Sapporo, Hokkaido, Japan.
[Ti] Título:Image findings of bisphosphonate related osteonecrosis of jaws comparing with osteoradionecrosis.
[So] Fuente:Dentomaxillofac Radiol;46(5):20160281, 2017 Jul.
[Is] ISSN:0250-832X
[Cp] País de publicación:England
[La] Idioma:eng
[Ab] Resumen:OBJECTIVES: The aim of this study was to evaluate image characteristics of bisphosphonate-related osteonecrosis of the jaws (BRONJ) and compare these with osteoradionecrosis (ORN). METHODS: 34 patients with BRONJ and 16 patients with ORN were included in this study. We investigated the CT and dental panoramic radiograph (DPR) images for osteolysis, osteosclerosis, sequestration, periosteal reaction, pathological fracture and spread of soft tissue inflammation around the jaws. RESULTS: Osteolysis, osteosclerosis, sequestration and spread of soft tissue inflammation around the jaws were common radiological features in both BRONJ and ORN. Osteolysis and spreading of soft tissue inflammation around the jaws were predominant in ORN, and by contrast osteosclerosis was predominant in BRONJ. Periosteal reaction was established in 15 of the 34 BRONJ cases, but none in the ORN cases. Pathological fractures were observed in 6 of 16 ORN cases, but none in BRONJ cases. CT was better for detection than DPR for osteolysis, osteosclerosis, sequestration and periosteal reactions. CONCLUSIONS: Image findings of BRONJ were characterized as a severe sclerotic change combined with osteolysis, sequestration, periosteal reaction and spread of soft tissue inflammation around the jaws.
[Mh] Términos MeSH primario: Osteonecrosis de los Maxilares Asociada a Difosfonatos/diagnóstico por imagen
Osteorradionecrosis/diagnóstico por imagen
Tomografía Computarizada por Rayos X/métodos
[Mh] Términos MeSH secundario: Adulto
Anciano
Anciano de 80 o más Años
Osteonecrosis de los Maxilares Asociada a Difosfonatos/patología
Femenino
Humanos
Masculino
Mediana Edad
Osteorradionecrosis/patología
Radiografía Panorámica
[Pt] Tipo de publicación:COMPARATIVE STUDY; JOURNAL ARTICLE
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171010
[Lr] Fecha última revisión:171010
[Sb] Subgrupo de revista:D
[Da] Fecha de ingreso para procesamiento:170210
[St] Status:MEDLINE
[do] DOI:10.1259/dmfr.20160281


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[PMID]:28980057
[Au] Autor:Zustin J; Amling M; Crazzolara R; Butscheidt S; Schulz A; Oheim R
[Ad] Dirección:Gemeinschaftspraxis für Pathologie, Pathologie-Hamburg, Lademannbogen 61-63, 22339, Hamburg, Deutschland. jozefzustin@t-online.de.
[Ti] Título:[Morphological characteristics of osteopetrosis].
[Ti] Título:Morphologische Veränderungen des Knochengewebes bei Osteopetrose..
[So] Fuente:Pathologe;, 2017 Oct 04.
[Is] ISSN:1432-1963
[Cp] País de publicación:Germany
[La] Idioma:ger
[Ab] Resumen:Osteopetrosis is a rare inherited bone disorder characterized by increased bone density owing to failure in bone resorption by the osteoclasts. The disease is genetically and histologically heterogeneous with a wide spectrum of microscopic findings. The histology varies from cases with a total absence of osteoclasts to bone biopsies characterized by high numbers of enlarged multinucleated osteoclasts on a background of sclerotic cancellous bone with or without additional defect of mineralization of the bone matrix. Here we present typical cases of human osteopetrosis on the basis of bone biopsies with four distinct genotypes (mutations of TNFRSF11A, TCIRG1, CNCL7, KINDLIN-3 genes) and discuss genotype-phenotype relationships. Analyzing human bone biopsies of rare skeletal disorders might improve our understanding of bone metabolism with possible implications for the clinical management of other bone diseases.
[Pt] Tipo de publicación:ENGLISH ABSTRACT; JOURNAL ARTICLE; REVIEW
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171005
[Lr] Fecha última revisión:171005
[St] Status:Publisher
[do] DOI:10.1007/s00292-017-0370-1


  10 / 4634 MEDLINE  
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[PMID]:28974398
[Au] Autor:Inoue A; Arai Y; Nakagawa S; Inoue H; Yoshihara Y; Yamazoe S; Kubo T
[Ad] Dirección:Department of Orthopaedics, Japanese Red Cross Kyoto Daiichi Hospital, Kyoto Japan; Department of Orthopaedics, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto Japan.
[Ti] Título:Differences in patellofemoral alignment as a result of patellar shape in cruciate-retaining total knee arthroplasty without patellar resurfacing at a minimum three-year follow-up.
[So] Fuente:Knee;, 2017 Sep 30.
[Is] ISSN:1873-5800
[Cp] País de publicación:Netherlands
[La] Idioma:eng
[Ab] Resumen:BACKGROUND: Although the patellofemoral (PF) joint shape of the femoral implant in a particular prosthetic design is identical, the articular surface shape of the patella varies between individuals. The present study investigated the effects of patellar shape on the postoperative PF joint in a cruciate-retaining total knee arthroplasty (CR-TKA) without patellar resurfacing and with at least three years of follow-up. METHODS: This study investigated 76 knees of 62 patients who underwent CR-TKA without patellar resurfacing for varus osteoarthritis. Shape of the patella was classified in terms of the patellar facet angle (angle formed by the medial and lateral articular surfaces) as measured on axial plain X-ray. Subjects were divided into Group A with patellar facet angle ≤126° (34 knees) and Group B with patellar facet angle >126° (42 knees). RESULTS: Anterior knee pain (AKP) was evident in four knees in Group A and one knee in Group B. Postoperative lateral patellar tilt angle was significantly larger in Group A than in Group B. Progression of osteosclerosis in the patella was present in 12 knees in Group A and three knees in Group B. CONCLUSION: This study found that for a patella with a small patellar facet angle, lateral tilt of the patella was significantly increased after TKA, and a high rate of osteosclerosis was evident at >3years after operation. Performing TKA without patellar resurfacing on a patella with a small patellar facet angle may entail a higher risk of postoperative AKP; pre-operative evaluation of the shape of the patella is therefore important.
[Pt] Tipo de publicación:JOURNAL ARTICLE
[Em] Mes de ingreso:1710
[Cu] Fecha actualización por clase:171004
[Lr] Fecha última revisión:171004
[St] Status:Publisher



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