The Gorham-Stout syndrome (Gorham's massive osteolysis)

Möller, Gertrude M.; Priemel, Matthias; Amling, Michael; Werner, Michael; Kuhlmey, Antje; Delling, G.

doi:10.1302/0301-620x.81b3.9468

Cited by 127 publications

(45 citation statements)

References 29 publications

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“…Gorham's disease is a rare disorder that may affect any part of the bone, but most commonly involves the skull, rib, pelvic girdle, shoulder 678. Surrounding soft tissues can also be involved 8.…”

Section: Discussionmentioning

confidence: 99%

“…There is no gender preference and genetic heredity cause. The diagnosis is based on combined clinical, radiological and histopathology findings after excluding neoplastic, immunologic, endocrinologic, infectious, and inflammatory diseases 6. However, the diagnosis is difficult and is often recognized retrospectively 28.…”

Section: Discussionmentioning

confidence: 99%

“…In the final stage, the remaining bone is reabsorbed and finally replaced by fibrous tissue. In the long bones, there is noted progress with concentric shrinkage and atrophy and tapering of the residual bone giving the appearance of “licked candy stick” deformity 6. Three-phase bone scintigraphy, demonstrated slightly decreased activity in the arterial phase, slightly increased activity in the blood pool phase, and increased activity in the delayed phase with normal thallium activity 11.…”

Section: Discussionmentioning

confidence: 99%

“…Möller et al . suggested hyperactive osteoclastic bone resorption causing osteolytic changes6 and that interleukin-6 (IL-6) may play a role stimulate osteoclast activity and increase sensitivity of osteoclast precursors 14. However, some authors suggested that the osteoclastosis” was not necessary and lymphangiogenesis or secondary to angiomatosis may be cause of osteolysis 1315…”

Section: Discussionmentioning

confidence: 99%

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Gorham’s disease in humerus treated with autogenous vascularized fibular graft

Kim

Kang

Kim³

et al. 2017

IJOO

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Gorham's disease is a rare disorder of the bone characterized by progressive massive osteolysis. The pathophysiology is unknown, and diagnosis is often difficult. Most cases are often recognized retrospectively. There is no standardized treatment and management for Gorham's disease. We report a case of an 18-year-old male presenting with a pathologic fracture in the humerus shaft diagnosed with Gorham's disease. Patient was treated with autogenous vascularized fibular graft with wide excision and a 10 years followup after first surgery.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

Gorham’s disease in humerus treated with autogenous vascularized fibular graft

Kim

Kang

Kim³

et al. 2017

IJOO

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“…Based on histopathological features, fibro-osseous disease in mice has some similarity to human Gorham-Stout Syndrome or Gorham’s massive osteolysis/angiomatosis (Möller et al, 1999; van der Linden-van der Zwaag and Onvlee, 2006). This rare human disease is usually discovered during radiographic evaluation of pathologic fracture.…”

Section: Discussionmentioning

confidence: 99%

Genetic determinants of fibro-osseous lesions in aged inbred mice

Berndt

Ackert‐Bicknell

Silva

et al. 2016

Experimental and Molecular Pathology

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Fibro-osseous lesions in mice are progressive aging changes in which the bone marrow is replaced to various degrees by fibrovascular stroma and bony trabeculae in a wide variety of bones. The frequency and severity varied greatly among 28 different inbred mouse stains, predominantly affecting females, ranging from 0% for 10 strains to 100% for KK/HlJ and NZW/LacJ female mice. Few lesions were observed in male mice and for 23 of the strains, no lesions were observed in males for any of the cohorts. There were no significant correlations between strain-specific severities of fibro-osseous lesions and ovarian (r=0.11; P=0.57) or endometrial (r=0.03; P=0.89) cyst formation frequency or abnormalities in parathyroid glands. Frequency of fibro-osseous lesions was most strongly associated (P < 10−6) with genome variations on chromosome (Chr) 8 at 90.6 and 90.8 Mb (rs33108071, rs33500669; P = 5.0 · 10−10, 1.3 · 10−6), Chr 15 at 23.6 and 23.8 Mb (rs32087871, rs45770368; P = 7.3 · 10−7, 2.7 · 10−6), and Chr 19 at 33.2, 33.4, and 33.6 Mb (rs311004232, rs30524929, rs30448815; P=2.8 · 10−6, 2.8 · 10−6, 2.8 · 10−6) in genome-wide association studies (GWAS). The relatively large number of candidate genes identified in the GWAS analyses suggests that this may be an extremely complex polygenic disease. These results indicate that fibro-osseous lesions are surprisingly common in many inbred strains of laboratory mice as they age. While this presents little problem in most studies that utilize young animals, it may complicate aging studies, particularly those focused on bone.

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Vascular Malformations

Boon

Vikkula

2019

Harper's Textbook of Pediatric Dermatology

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The Gorham-Stout syndrome (Gorham's massive osteolysis)

Cited by 127 publications

References 29 publications

Gorham’s disease in humerus treated with autogenous vascularized fibular graft

Gorham’s disease in humerus treated with autogenous vascularized fibular graft

Genetic determinants of fibro-osseous lesions in aged inbred mice

Vascular Malformations

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