Polyostotic Fibrous Dysplasia And Acromegaly

Scurry, Murphy T.

doi:10.1001/archinte.1964.03860070086008

Cited by 44 publications

(7 citation statements)

References 12 publications

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“…Scurry et al [14] have described polyostotic fibrous dysplasia and acromegaly in a 38-year-old woman with elevated level of serum growth hormone, while Derome and Visot have described a 26-year-old woman with monostotic fibrous dysplasia of the skull and acromegaly [15]. Harris has also described a case of incomplete Albright's syndrome in combination with elevated growth hormone and prolactin, and has cited three cases from the literature [161.…”

Section: Discussionmentioning

confidence: 99%

Fibrous dysplasia of the skull with acromegaly and sarcomatous transformation

1989

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Two cases of fibrous dysplasia of the skull are reported. Both patients were young women with acromegaly and were treated with radiotherapy. Progressive pareses of cranial nerves, pain, and a malignant course of the disease were characteristic in both patients, and the diagnosis of osteogenous sarcoma proved in one of them by histological examination. The clinical picture of fibrous dysplasia of the skull and the role of radiotherapy with the risk of development of malignancy is discussed.

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Section: Discussionmentioning

confidence: 99%

Fibrous dysplasia of the skull with acromegaly and sarcomatous transformation

1989

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“…The first well documented case of hypersomatotropinism in a patient with MAS was published by Scurry et al 59 . To date there are more than 50 cases reported in the literature.…”

Section: Hypersomatotropinismmentioning

confidence: 99%

McCune-Albright Syndrome and Disorders Due to Activating Mutations of GNAS1

Díaz¹,

Danon²,

Crawford³

2007

Journal of Pediatric Endocrinology and Metabolism

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It has been more than seven decades since Drs. Fuller Albright and Donovan McCune published the first reports on individuals with McCune-Albright syndrome (MAS). Since then, the classic triad of precocious puberty, café-aulait spots, and polyostotic bone dysplasia continues to define the syndrome. However, having gathered a better picture of the pathophysiology of MAS, the way this condition is understood has changed. Isolated activating mutations of the alpha subunit of the G protein (GNAS1) have been found in different tissues, including pituitary adenomas, thyroid adenomas, ovarian cysts, monostotic bone dysplasia, and the adrenal glands, to name a few. For this reason, we have added 'and disorders due to activating mutations of GNAS1' to the title of this review. We discuss here the clinical consequences of GNAS1 activating mutations in different body systems and organs, the diagnostic approach to MAS, and the current therapeutic recommendations.

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“…Purely endocrine features described in MAS include non-autoimmune hyperthyroidism, acromegaly, Cushing syndrome, hyperprolactinemia, and hyperparathyroidism. Bony problems include polyostotic fibrous dysplasia [3-7]. Several non-endocrine features more recently attributed to the molecular defect include platelet abnormalities, hepatobiliary disease, cardiac hypertrophy, and sudden or premature death [2,8-11].…”

Section: Introductionmentioning

confidence: 99%

Allergic manifestations and cutaneous histamine responses in patients with McCune Albright syndrome

Jacobson¹,

Turpin²,

Sands³

2013

World Allergy Organization Journal

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BackgroundMcCune Albright syndrome (MAS) is a rare disorder characterized by precocious puberty, café-au-lait spots, and fibrous dysplasia. Its cause is an activating mutation in the GNAS gene, encoding a subunit of the stimulatory G protein, Gsalpha (Gsα). The action of any mediator that signals via Gsα and cyclic AMP can be up regulated in MAS. We had observed gastritis, gastroesophageal reflux, and anaphylaxis in McCune Albright patients.ObjectiveAs histamine is known to signal via histamine 1 (H1) and histamine 2 (H2) receptors, which couple with stimulatory G proteins, we attempted to mechanistically link histamine responsiveness to the activating GNAS mutation. We hypothesized that responsiveness to histamine skin testing would differ between MAS patients and healthy controls.Patients and methodsAfter obtaining informed consent, we performed a systematic review of histamine responsiveness and allergic manifestations in 11 MAS patients and 11 sex-matched, Tanner-stage matched controls. We performed skin prick testing, quantifying the orthogonal diameters of wheals and erythema. We also quantitated G protein mRNA expression.ResultsThe peak wheal and flare responses to histamine were significantly higher in MAS patients compared to controls.ConclusionsThis study suggests that MAS patients may be at risk for exaggerated histamine responsiveness compared to unaffected controls.

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Polyostotic Fibrous Dysplasia And Acromegaly

Cited by 44 publications

References 12 publications

Fibrous dysplasia of the skull with acromegaly and sarcomatous transformation

Fibrous dysplasia of the skull with acromegaly and sarcomatous transformation

McCune-Albright Syndrome and Disorders Due to Activating Mutations of GNAS1

Allergic manifestations and cutaneous histamine responses in patients with McCune Albright syndrome

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