Platelet-derived growth factor receptor-β in Gorham's disease

Hagendoorn, Jeroen; Padera, Timothy P.; Yock, Torunn I.; Nielsen, G. Petur; Tomaso, Emmanuelle di; Duda, Dan G.; DeLaney, Thomas F.; Gaissert, Henning A.; Pearce, Jennifer M.; Rosenberg, Andrew E.; Jain, Rakesh K.; Ebb, David H.

doi:10.1038/ncponc0660

Cited by 63 publications

(80 citation statements)

References 16 publications

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“…It is controversial whether osteoclasts are present or not [6]. In some cases, such as in those of Gorham and Stout [3], osteoclasts have not been found in areas of resorption or reparative osteogenesis, and therefore angiomatosis is thought to be responsible for the process [7,8]. Recent studies have suggested that vascular endothelial growth factor is a marker of disease activity [9].…”

Section: Discussionmentioning

confidence: 99%

Gorham-Stout Disease and Cerebrospinal Fluid Otorrhea

Hernández-Marqués

González²,

Ortega³

et al. 2011

Pediatr Neurosurg

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Objective and Importance: Gorham-Stout disease is a rare entity characterized by vascular proliferation causing local destruction of bone tissue. Owing to its low incidence and variable clinical presentation, the diagnosis requires a high degree of awareness by the clinician. Clinical Presentation: We present the case of a 2-year-old boy diagnosed of Gorham-Stout syndrome with involvement of the temporal bone and secondary cerebrospinal fluid (CSF) leakage. Intervention: Because of the CSF leakage, the patient required two surgical interventions. The second intervention included mastectomy and placement of a patch and a lumbar drainage device during 50 days, after which the leakage ceased. Conclusion: Gorham-Stout disease is a rare condition that can affect the skull base and even present with CSF leakage.

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Section: Discussionmentioning

confidence: 99%

Gorham-Stout Disease and Cerebrospinal Fluid Otorrhea

Hernández-Marqués

González²,

Ortega³

et al. 2011

Pediatr Neurosurg

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“…С учетом вариабельности скорости остеолиза наиболее перспективными биомаркерами его активности счита-ются VGEF-A [7,10,37] и интерлей-кин-6 [12,17,18,43], уровень которых при GSD может быть повышен. Вме-сте с тем показатели этих цитокинов могут оставаться и нормальными [25,37], поэтому продолжаются активные поиски других биомаркеров остеоли-за -VEGF-C [37], PDGF-BB [25], sRANKL и остеопротегерина [18].…”

Section: рисunclassified

A Rare Variant of Angiogenic Vertebral Body Destruction in a Child: Gorham’s Disease?

Мушкин¹,

Евсеев²,

Николаев³

et al. 2018

Hir. pozvonoč.

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“…Another important point about the histopathology of Gorham-Stout syndrome was noted by Colucci et al [11] (in 2006), who found that the cells they isolated from a patient's lesion belonged to a monocyte-macrophage lineage and could release high amounts of osteoclastogenic and angiogenic molecules. Additionally, Hagendoorn et al [12] (in 2006) underlined the critical role that could play the signaling pathway of the PDGFR-b (receptor of the lymphangiogenic growth factor Platelet Derived Growth Factor BB) in the pathogenetic mechanism of the disease. Besides, in 2007, Bruch-Gerharz et al [5] tried to shed more light onto the pathogenesis of the syndrome, writing about lymphatic vascular malformations involving the skin and the soft tissues adjacent to the diseased bone.…”

Section: Etiopathologymentioning

confidence: 99%

Vanishing bone disease (Gorham-Stout syndrome): A review of a rare entity

Nikolaou¹

2014

WJO

102

104

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Vanishing bone disease (Gorham-Stout syndrome) is a rare entity of unknown etiology, characterized by destruction of osseous matrix and proliferation of vascular structures, resulting in destruction and absorption of bone. Despite the extensive investigation of the pathogenetic mechanisms of the disease, its etiology hasn't been clarified and several theories exist. The syndrome can affect one or multiple bones of the patient, including the skull, the upper and lower extremities, the spine and pelvis. The clinical presentation of a patient suffering from vanishing bone disease includes, pain, functional impairment and swelling of the affected region, although asymptomatic cases have been reported, as well as cases in which the diagnosis was made after a pathologic fracture. In this short review we summarize the theories regarding the etiology as well as the clinical presentation, the diagnostic approach and treatment options of this rare disease.© 2014 Baishideng Publishing Group Inc. All rights reserved. Key words:Vanishing bone disease; Gorham-Stout syndrome; Histology; Diagnosis; Treatment Core tip: Vanishing bone disease (Gorham-Stout syndrome) is a rare entity of unknown etiology, characterized by destruction of osseous matrix and proliferation of vascular structures, resulting in destruction and absorption of bone. The syndrome can affect one or multiple bones of the patient, including the skull, the upper and lower extremities, the spine and pelvis. Physicians should be aware of the existence of this rare entity and reliably direct affected patients to right diagnosis and therapeutic approach.

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Platelet-derived growth factor receptor-β in Gorham's disease

Cited by 63 publications

References 16 publications

Gorham-Stout Disease and Cerebrospinal Fluid Otorrhea

Gorham-Stout Disease and Cerebrospinal Fluid Otorrhea

A Rare Variant of Angiogenic Vertebral Body Destruction in a Child: Gorham’s Disease?

Vanishing bone disease (Gorham-Stout syndrome): A review of a rare entity

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