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[PMID]:29049202
[Au] Autor:Liu M; Liu W; Qiao C; Han B
[Ad] Endereço:aDepartment of Oral and Maxillofacial Surgery bJilin Provincial Key Laboratory of Tooth Development and Bone Remodeling cDepartment of Oral Pathology, School and Hospital of Stomatology, Jilin University, Changchun, China.
[Ti] Título:Mandibular Gorham-Stout disease: A case report and literature review.
[So] Source:Medicine (Baltimore);96(42):e8184, 2017 Oct.
[Is] ISSN:1536-5964
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:RATIONALE: Gorham-Stout disease (GSD) is characterized by aggressive bone resorption, proliferation of vascular or lymphatic vessels, and soft-tissue swelling. Bones that initially appear normal start to resorb, partially or completely. However, the etiology of GSD is unknown. PATIENT CONCERNS: A 29-year-old man with a chief complaint of toothache and mobility in the lower right mandible for the previous 1 year. DIAGNOSES: Gorham-Stout disease (GSD). INTERVENTIONS: The RANK-ligand inhibitor denosumab was suggested to use to inhibit the development of osteoclasts and slow mandibular resorption. In addition, we proposed resection of the remaining mandible and reconstruction via vascularized bone graft, after resorption of the mandible had become stationary. OUTCOMES: Regular follow-ups were advised to this patient to monitor the stability of bone resorption prior to any surgical intervention. LESSONS: We strongly recommend that every attempt should be made for early diagnosis and prompt effective medical and surgical management. The failure to do so results in further complications and poor prognosis.
[Mh] Termos MeSH primário: Doenças Mandibulares/complicações
Osteólise Essencial/complicações
Odontalgia/etiologia
[Mh] Termos MeSH secundário: Adulto
Seres Humanos
Masculino
Mandíbula/fisiopatologia
Doenças Mandibulares/fisiopatologia
Osteólise Essencial/fisiopatologia
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE; REVIEW
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171122
[Lr] Data última revisão:
171122
[Sb] Subgrupo de revista:AIM; IM
[Da] Data de entrada para processamento:171020
[St] Status:MEDLINE
[do] DOI:10.1097/MD.0000000000008184


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[PMID]:28009802
[Au] Autor:Tan E; T Mehlman C; Baker M
[Ad] Endereço:*Department of Orthopaedic Surgery, Grandview Hospital & Medical Center, Ohio University Heritage College of Osteopathic Medicine, Dayton, OH †Cincinnati Children's Hospital Medical Center, Cincinnati, OH.
[Ti] Título:Benign Osteolytic Lesions in Children With Previously Normal Radiographs.
[So] Source:J Pediatr Orthop;37(4):e282-e285, 2017 Jun.
[Is] ISSN:1539-2570
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:BACKGROUND: Benign bone tumors are common in children and are usually diagnosed incidentally or after pathologic fractures. However, case reports of children with lytic lesions with previously normal radiographs are rare. This series presents several such cases. METHODS: This was a retrospective case series of 8 benign osteolytic lesions in 8 pediatric patients (5 female and 3 males). All have previous radiographs of the effected extremity showing no pathology. All participants were at a single institution. RESULTS: Eight cases were identified with 2 aneurysmal bone cysts and 6 nonossifying fibromas. Average age at discovery was 10.75 years old. The range of time between normal and pathologic radiographs was from 2.5 to 7.25 years. Both cases of aneurysmal bone cysts were histologically confirmed and treated with curettage and grafting with one recurrence. One of 6 nonossifying fibromas was treated surgically with the remainder treated with casting for pathologic fractures or observation for incidentally identified lesions. CONCLUSIONS: Benign osteolytic lesions such as aneurysmal bone cysts and nonossifying fibromas can arise in a short time period. The average time between normal and pathologic radiographs was 4.6 years. Prelesion radiographs were obtained to evaluate traumas and leg length discrepancies. CLINICAL RELEVANCE: Radiographs taken before diagnosis in children with osseous lesions are rare, but likely under recognized. Further studies of such instances in larger databases may provide a noninvasive means to reach diagnostic or etiologic conclusions.
[Mh] Termos MeSH primário: Cistos Ósseos Aneurismáticos/diagnóstico por imagem
Neoplasias Ósseas/diagnóstico por imagem
Fibroma/diagnóstico por imagem
Osteólise Essencial/diagnóstico por imagem
[Mh] Termos MeSH secundário: Adolescente
Cistos Ósseos Aneurismáticos/patologia
Neoplasias Ósseas/patologia
Criança
Pré-Escolar
Curetagem
Feminino
Fibroma/patologia
Fraturas Espontâneas/diagnóstico por imagem
Seres Humanos
Masculino
Osteólise Essencial/patologia
Radiografia
Estudos Retrospectivos
Fatores de Tempo
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Em] Mês de entrada:1709
[Cu] Atualização por classe:170912
[Lr] Data última revisão:
170912
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161224
[St] Status:MEDLINE
[do] DOI:10.1097/BPO.0000000000000918


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[PMID]:28004375
[Au] Autor:Franco-Barrera MJ; Zavala-Cerna MG; Aguilar-Portillo G; Sánchez-Gomez DB; Torres-Bugarin O; Franco-Barrera MA; Roa-Encarnacion CM
[Ad] Endereço:Division de Cirugia Maxilofacial, Hospital Universitario de Puebla, Benemerita Universidad de Puebla, Puebla, Puebla, Mexico.
[Ti] Título:Gorham-Stout Disease: a Clinical Case Report and Immunological Mechanisms in Bone Erosion.
[So] Source:Clin Rev Allergy Immunol;52(1):125-132, 2017 Feb.
[Is] ISSN:1559-0267
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:Gorham-Stout disease (GSD) is a rare condition of osteolysis with excessive lymphangiogenesis within bone tissue. The etiology of this condition remains unknown but seems to affect mainly children and young adults of both genders all over the world. Unfortunately, there is no standardized method for diagnosis; however, histopathology remains as the gold standard. This condition is often misdiagnosed due to its varying clinical presentations from case-to-case. Here, we report the case of an 8-year-old girl who presented with chronic mandibular pain during mastication and received multiple antibiotic treatment due to infectious origin suspicion. After integrating information from clinical manifestations, radiographic, laboratory, and histopathology information, she was diagnosed with GSD. Additionally, due to the lack of literature with respect to insights into biological mechanisms and standardized treatment for this condition, we underwent a literature revision to provide information related to activation of cells from the immune system, such as macrophages, T-cells, and dendritic cells, and their contribution to the lymphangiogenesis, angiogenesis, and osteoclastogenic process in GSD. It is important to consider these mechanisms in patients with GSD, especially since new studies performed in earlier stages are required to confirm their use as novel diagnostic tools and find new possibilities for treatment.
[Mh] Termos MeSH primário: Osteólise Essencial/patologia
[Mh] Termos MeSH secundário: Criança
Feminino
Seres Humanos
Mandíbula/anormalidades
Osteólise Essencial/diagnóstico
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE; REVIEW
[Em] Mês de entrada:1702
[Cu] Atualização por classe:171107
[Lr] Data última revisão:
171107
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161223
[St] Status:MEDLINE
[do] DOI:10.1007/s12016-016-8594-z


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[PMID]:27829197
[Au] Autor:Renacci RM; Bartolotta RJ
[Ad] Endereço:Georgetown University School of Medicine, 3900 Reservoir Road, NW, Washington, DC 20057. Electronic address: rmr92@georgetown.edu.
[Ti] Título:Gorham disease: lymphangiomatosis with massive osteolysis.
[So] Source:Clin Imaging;41:83-85, 2017 Jan - Feb.
[Is] ISSN:1873-4499
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:Gorham disease (also referred to as vanishing bone disease or idiopathic massive osteolysis) is a rare skeletal condition that results in the localized replacement of bone with angiomas and fibrosis. The etiology and most effective treatment for this nonhereditary condition remain controversial in the medical literature. To our knowledge, we present the first case of post-traumatic Gorham disease that includes MR imaging (before and after radiation therapy), post-radiation CT with evidence of treatment effect (sclerosis), as well as asymptomatic bilateral renal lymphangiomata.
[Mh] Termos MeSH primário: Nefropatias/complicações
Nefropatias/diagnóstico por imagem
Linfangioma/complicações
Linfangioma/diagnóstico por imagem
Osteólise Essencial/complicações
Osteólise Essencial/diagnóstico por imagem
[Mh] Termos MeSH secundário: Adulto
Seres Humanos
Imagem por Ressonância Magnética
Masculino
Osteólise Essencial/radioterapia
Tomografia Computadorizada por Raios X
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Em] Mês de entrada:1701
[Cu] Atualização por classe:170112
[Lr] Data última revisão:
170112
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161110
[St] Status:MEDLINE


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[PMID]:27582313
[Au] Autor:Seo GS; Dieudonne G; Mooney SA; Monu JU
[Ad] Endereço:1 University of Rochester, Rochester, New York, USA.
[Ti] Título:Unexplained "massive osteolysis of femoral head" (MOFH) after acetabular fracture: occurrence and suggested patho-etiology.
[So] Source:Acta Radiol;58(6):710-718, 2017 Jun.
[Is] ISSN:1600-0455
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:Background We observed cases of extensive osteolysis of the femoral head following acetabular fractures even though the original fracture did not involve the femoral head or neck. This observation has been called massive osteolysis of the femoral head (MOFH). Purpose To evaluate the clinical and imaging features of MOFH to gain a better understanding of its patho-etiology. Material and Methods We retrospectively collected cases of acetabular fractures seen over a period of 10 years and evaluated the clinical features and imaging characteristics. The findings were compared with the features of other complications such as osteonecrosis, rapidly progressive osteoarthritis, or post-traumatic osteolysis. Results Fifteen patients (M:F ratio 9:6; mean age, 61.6 years) out of 244 had MOFH (prevalence: 6.1%). Motor vehicle collision and falls were the most common mechanisms of injury. The time interval for developing MOFH was in the range of 1-18 months after the initial injury. Patterns of femoral head osteolysis varied from eccentric (12 cases) to transcervical (3 cases). Four cases of eccentric osteolysis developed high-degree of osteolysis. MOFH was observed near the surgical hardware in 6/15 cases. One biopsy specimen did not reveal typical features of osteoarthritis or avascular necrosis. Conclusion MOFH appears to be a distinct entity from avascular necrosis or rapidly progressive osteoarthritis. It is suggested that MOFH is a variant of post-traumatic osteolysis that is evident in a subset of patients.
[Mh] Termos MeSH primário: Acetábulo/lesões
Cabeça do Fêmur
Fraturas Ósseas/complicações
Osteólise Essencial/etiologia
[Mh] Termos MeSH secundário: Adolescente
Adulto
Idoso
Idoso de 80 Anos ou mais
Feminino
Seres Humanos
Masculino
Meia-Idade
Estudos Retrospectivos
Adulto Jovem
[Pt] Tipo de publicação:COMPARATIVE STUDY; JOURNAL ARTICLE
[Em] Mês de entrada:1706
[Cu] Atualização por classe:170607
[Lr] Data última revisão:
170607
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:160902
[St] Status:MEDLINE
[do] DOI:10.1177/0284185116665422


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[PMID]:28348444
[Au] Autor:García V; Alonso-Claudio G; Gómez-Hernández MT; Chamorro AJ
[Ad] Endereço:Departamento de Medicina Interna. Hospital Clínico Universitario de Salamanca. Salamanca. España.
[Ti] Título:Sirolimus on Gorham-Stout disease. Case report.
[So] Source:Colomb Med (Cali);47(4):213-216, 2016 Dec 30.
[Is] ISSN:1657-9534
[Cp] País de publicação:Colombia
[La] Idioma:eng
[Ab] Resumo:BACKGROUND: Gorham-Stout disease (GSD) is a rare disease of unknown etiology characterized by vascular proliferation that produces destruction of bone matrix. CASE DESCRIPTION: This case is about 43 year old woman who begins with pain in sternum, dyspnea, abdominal mass and, serous-hematic pleural effusion. Imaging tests were performed showing lesions on 6 and 10 left ribs archs. Later, a thoracotomy was performed observed absence of the end of the 6 and lung, pleural and costal biopsy was token. The histologic features described lymphatic vascular proliferation in bone tissue of chest wall. Other pathologies were excluded and in view of the findings, GSD diagnosis was made. TREATMENT AND OUTCOME: treatment was initiated with sirolimus achieving remission of the disease after the first month; however, because the presence of metrorrhagia the treatment was discontinued, reappearing symptoms afterwards. For that reason the treatment was restarted getting disappearance of the symptoms again, 4 weeks later. CLINICAL RELEVANCE: we present the first clinical cases of EGS with pleural effusion with response to sirolimus treatment that could be an alternative to the current therapy.
[Mh] Termos MeSH primário: Imunossupressores/uso terapêutico
Osteólise Essencial/tratamento farmacológico
Sirolimo/uso terapêutico
[Mh] Termos MeSH secundário: Adulto
Feminino
Seres Humanos
Osteólise Essencial/diagnóstico
Osteólise Essencial/fisiopatologia
Derrame Pleural/tratamento farmacológico
Derrame Pleural/etiologia
Resultado do Tratamento
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Immunosuppressive Agents); W36ZG6FT64 (Sirolimus)
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171023
[Lr] Data última revisão:
171023
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:170329
[St] Status:MEDLINE


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[PMID]:27648707
[Au] Autor:Papadakis GZ; Millo C; Bagci U; Blau J; Collins MT
[Ad] Endereço:From the *Radiology and Imaging Sciences, Warren Grant Magnuson Clinical Center, National Institutes of Health, Bethesda; †Division of Nuclear Medicine, RAD&IS, Clinical Center, National Institutes of Health, Bethesda, MD; ‡Center for Research in Computer Vision, Electrical and Computer Science Department, University of Central Florida, Orlando, FL; §National Institute of Child Health and Human Development, National Institutes of Health, Bethesda; and ∥Craniofacial and Skeletal Diseases Branch, National Institutes of Dental and Craniofacial Research, Bethesda, MD.
[Ti] Título:18F-NaF and 18F-FDG PET/CT in Gorham-Stout Disease.
[So] Source:Clin Nucl Med;41(11):884-885, 2016 Nov.
[Is] ISSN:1536-0229
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:Gorham-Stout disease (GSD) is an extremely rare skeletal disorder of unknown etiology characterized by benign proliferation of vascular or lymphatic channels, leading to progressive bone resorption. We report on a patient diagnosed with GSD affecting the right scapula and the right ribs, who underwent PET/CT scans using F-FDG and F-NaF. The remnant upper portion of the affected scapula did not show F-FDG uptake but demonstrated markedly increased F-NaF activity. Furthermore, intense F-NaF activity was seen on the right posterior ribs, which were actively being resorbed, suggesting the potential application of F-NaF-PET/CT imaging in GSD diagnosis and follow-up.
[Mh] Termos MeSH primário: Osteólise Essencial/diagnóstico por imagem
Tomografia Computadorizada com Tomografia por Emissão de Pósitrons
[Mh] Termos MeSH secundário: Adulto
Radioisótopos de Flúor
Fluordesoxiglucose F18
Seres Humanos
Masculino
Compostos Radiofarmacêuticos
Fluoreto de Sódio
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Fluorine Radioisotopes); 0 (Radiopharmaceuticals); 0Z5B2CJX4D (Fluorodeoxyglucose F18); 8ZYQ1474W7 (Sodium Fluoride)
[Em] Mês de entrada:1612
[Cu] Atualização por classe:171101
[Lr] Data última revisão:
171101
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:160921
[St] Status:MEDLINE


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[PMID]:27514588
[Au] Autor:Itkin M; McCormack FX
[Ad] Endereço:Interventional Radiology, Hospital of the University of Pennsylvania, Penn Medicine, 1 Silverstein, 3400 Spruce Street, Philadelphia, PA 19104, USA. Electronic address: itkinmax@uphs.upenn.edu.
[Ti] Título:Nonmalignant Adult Thoracic Lymphatic Disorders.
[So] Source:Clin Chest Med;37(3):409-20, 2016 09.
[Is] ISSN:1557-8216
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:The thoracic lymphatic disorders are a heterogeneous group of uncommon conditions that are associated with thoracic masses, interstitial pulmonary infiltrates, and chylous complications. Accurate diagnosis of the thoracic lymphatic disorders has important implications for the newest approaches to management, including embolization and treatment with antilymphangiogenic drugs. New imaging techniques to characterize lymphatic flow, such as dynamic contrast-enhanced magnetic resonance lymphangiogram, are redefining approaches to disease classification and therapy.
[Mh] Termos MeSH primário: Pneumopatias/congênito
Neoplasias Pulmonares
Linfangiectasia/congênito
Linfangioleiomiomatose
Anormalidades Linfáticas
Osteólise Essencial
Síndrome das Unhas Amareladas
[Mh] Termos MeSH secundário: Adulto
Seres Humanos
Linfangioma
Doenças Linfáticas
[Pt] Tipo de publicação:JOURNAL ARTICLE; REVIEW
[Em] Mês de entrada:1702
[Cu] Atualização por classe:170214
[Lr] Data última revisão:
170214
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:160813
[St] Status:MEDLINE


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[PMID]:27331246
[Au] Autor:Amirjamshidi A; Karimi-Yarandi K; Hosseini M; Ghazy-Mirsaeed S; Pourrashidi-Boshrabadi A
[Ad] Endereço:a Department of Neurosurgery , Sina Hospital, Tehran University of Medical Sciences , Tehran , Iran.
[Ti] Título:Painful sporadic osteolysis of the parietal bone 'Gorham's disease'.
[So] Source:Br J Neurosurg;30(6):687-688, 2016 Dec.
[Is] ISSN:1360-046X
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:We report a 62-year old man, with painful osteolysis 'Gorham disease' of skull. To ameliorate pain, large craniotomy and cranioplasty was performed. There was no sign of recurrence after one year. A brief review of the literature is presented.
[Mh] Termos MeSH primário: Procedimentos Neurocirúrgicos/métodos
Osteólise Essencial/cirurgia
[Mh] Termos MeSH secundário: Craniotomia
Seres Humanos
Imagem Tridimensional
Masculino
Meia-Idade
Osteólise
Osteólise Essencial/diagnóstico por imagem
Osso Parietal/diagnóstico por imagem
Osso Parietal/cirurgia
Tomografia por Emissão de Pósitrons
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Em] Mês de entrada:1703
[Cu] Atualização por classe:170817
[Lr] Data última revisão:
170817
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:160623
[St] Status:MEDLINE


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[PMID]:27102925
[Au] Autor:Gataa IS; Nader NH; Abdallah DT
[Ad] Endereço:Assistant Professor and Head, Department of Oral and Maxillofacial Surgery, School of Dentistry, University of Sulaimani, Sulaimani, Iraq. Electronic address: dribsg@yahoo.com.
[Ti] Título:Massive Craniofacial Gorham Disease Treated Successfully by Cisplatin and 5-Fluorouracil With Ten Years of Follow-Up: A Case Report and Literature Review.
[So] Source:J Oral Maxillofac Surg;74(9):1774-82, 2016 Sep.
[Is] ISSN:1531-5053
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:Gorham disease (GD) is a rare osteolytic condition of unknown etiology that causes spontaneous, progressive bone resorption. The maxillofacial area is one of the regions most frequently involved in this disease. GD is characterized by its aggressiveness and rarity; therefore, the treatment modalities remain controversial, and no specific treatment method has been proved effective. The present report describes a case of GD with massive craniofacial bone involvement that was treated effectively using a combination of 5-fluorouracil and cisplatin, with 10 years of follow-up.
[Mh] Termos MeSH primário: Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico
Cisplatino/administração & dosagem
Fluoruracila/administração & dosagem
Doenças Maxilomandibulares/tratamento farmacológico
Doenças Orbitárias/tratamento farmacológico
Osteólise Essencial/tratamento farmacológico
[Mh] Termos MeSH secundário: Adolescente
Biópsia
Feminino
Seguimentos
Seres Humanos
Doenças Maxilomandibulares/diagnóstico por imagem
Doenças Orbitárias/diagnóstico por imagem
Osteólise Essencial/diagnóstico por imagem
Radiografia Panorâmica
Crânio/diagnóstico por imagem
Tomografia Computadorizada por Raios X
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE; REVIEW
[Nm] Nome de substância:
Q20Q21Q62J (Cisplatin); U3P01618RT (Fluorouracil)
[Em] Mês de entrada:1704
[Cu] Atualização por classe:170817
[Lr] Data última revisão:
170817
[Sb] Subgrupo de revista:AIM; D; IM
[Da] Data de entrada para processamento:160423
[St] Status:MEDLINE



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