Sensitivity of patients with familial cerebral cavernous malformations to therapeutic radiation

Golden, Michael J.; Saeidi, Saba; Liem, Ben; Marchand, Éric; Morrison, Leslie; Hart, Blaine L.

doi:10.1111/1754-9485.12269

Cited by 13 publications

(6 citation statements)

References 4 publications

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“…However, small risks do accompany use of ionizing radiation as it may promote CCM formation, and CCM patients may need repeated imaging. 67 The suspicion of CCM on CT should be followed by MRI. 68 MRI is the imaging test of choice for detection and characterization of CCMs, with near-perfect sensitivity and great specificity.…”

Section: Imaging Ccms and Reporting Standardsmentioning

confidence: 99%

Synopsis of Guidelines for the Clinical Management of Cerebral Cavernous Malformations: Consensus Recommendations Based on Systematic Literature Review by the Angioma Alliance Scientific Advisory Board Clinical Experts Panel

et al. 2017

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BACKGROUND: Despite many publications about cerebral cavernous malformations (CCMs), controversy remains regarding diagnostic and management strategies. OBJECTIVE: To develop guidelines for CCM management. METHODS: The Angioma Alliance (www.angioma.org), the patient support group in the United States advocating on behalf of patients and research in CCM, convened a multidisciplinary writing group comprising expert CCM clinicians to help summarize the existing literature related to the clinical care of CCM, focusing on 5 topics: (1) epidemiology and natural history, (2) genetic testing and counseling, (3) diagnostic criteria and radiology standards, (4) neurosurgical considerations, and (5) neurological considerations. The group reviewed literature, rated evidence, developed recommendations, and established consensus, controversies, and knowledge gaps according to a prespecified protocol. RESULTS: Of 1270 publications published between January 1, 1983 and September 31, 2014, we selected 98 based on methodological criteria, and identified 38 additional recent or relevant publications. Topic authors used these publications to summarize current knowledge and arrive at 23 consensus management recommendations, which we rated by class (size of effect) and level (estimate of certainty) according to the American Heart Association/American Stroke Association criteria. No recommendation was level A (because of the absence of randomized controlled trials), 11 (48%) were level B, and 12 (52%) were level C. Recommendations were class I in 8 (35%), class II in 10 (43%), and class III in 5 (22%). CONCLUSION: Current evidence supports recommendations for the management of CCM, but their generally low levels and classes mandate further research to better inform clinical practice and update these recommendations. The complete recommendations document, including the criteria for selecting reference citations, a more detailed justification of the respective recommendations, and a summary of controversies and knowledge gaps, was similarly peer reviewed and is available on line www.angioma.org/CCMGuidelines.

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Section: Imaging Ccms and Reporting Standardsmentioning

confidence: 99%

Synopsis of Guidelines for the Clinical Management of Cerebral Cavernous Malformations: Consensus Recommendations Based on Systematic Literature Review by the Angioma Alliance Scientific Advisory Board Clinical Experts Panel

et al. 2017

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“…16 Stereotactic radiosurgery has been proposed, but there is controversy about its effectiveness and concern about complications and radiationinduced genesis of new CAs. 19,20 The rates of development of new lesions, and of first SH in asymptomatic CAs, are far too low to allow meaningful testing or to compel primary prevention strategies for CASH. Cases with recent SH, where surgical resection is not undertaken (mostly in deep and brainstem locations), are the most likely to be followed expectantly per current evidence-based guidelines, 10 with clinical equipoise for testing novel therapies aimed at preventing rebleeding.…”

Section: General Information Cavernous Angioma: a Common Lesion An Umentioning

confidence: 99%

Trial Readiness in Cavernous Angiomas With Symptomatic Hemorrhage (CASH)

et al. 2018

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“…Brain lesion count was not assessed in one subject, a 43-year-old woman with more than 200 lesions who had a history of radiation therapy for head and neck malignancy in addition to fCCM. Cranial radiation is known to increase the risk of sporadic CCM development (24), likely because of a “second-hit” mechanism in patients with fCCM mutations (23). Subjects with SFCs had significantly higher brain lesion counts than those without SFCs (proportional increase: 5.7; 95% confidence interval: 2.6, 12.4; P < .001) and also tended to have more lesions than expected for their age (proportional increase: 1.6; 95% confidence interval: 0.9, 3.1; P = .131).…”

Section: Resultsmentioning

confidence: 99%

“…An additional 12 patients with fCCM identified by means of a records search met the inclusion criteria, for a total of 38 patients with fCCM. Of the 38 patients, 24 have previously been reported on (22,23). Previous publications dealt with neuroimaging and genetic aspects of fCCM, whereas herein we report abdominal findings in these patients.…”

Section: Methodsmentioning

confidence: 99%

Familial Cerebral Cavernous Malformations Are Associated with Adrenal Calcifications on CT Scans: An Imaging Biomarker for a Hereditary Cerebrovascular Condition

et al. 2017

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Purpose To determine if adrenal calcifications seen at computed tomography (CT) are associated with familial cerebral cavernous malformations (fCCMs) in carriers of the CCM1 Common Hispanic Mutation. Materials and Methods This study was approved by the institutional review board. The authors retrospectively reviewed abdominal CT scans in 38 patients with fCCM, 38 unaffected age- and sex-matched control subjects, and 13 patients with sporadic, nonfamilial cerebral cavernous malformation (CCM). The size, number, and laterality of calcifications and the morphologic characteristics of the adrenal gland were recorded. Brain lesion count was recorded from brain magnetic resonance (MR) imaging in patients with fCCM. The prevalence of adrenal calcifications in patients with fCCM was compared with that in unaffected control subjects and those with sporadic CCM by using the Fisher exact test. Additional analyses were performed to determine whether age and brain lesion count were associated with adrenal findings in patients with fCCM. Results Small focal calcifications (SFCs) (≤5 mm) were seen in one or both adrenal glands in 19 of the 38 patients with fCCM (50%), compared with 0 of the 38 unaffected control subjects (P < .001) and 0 of the 13 subjects with sporadic CCM (P = .001). Adrenal calcifications in patients with fCCM were more frequently left sided, with 17 of 19 patients having more SFCs in the left adrenal gland than the right adrenal gland and 50 of the 61 observed SFCs (82%) found in the left adrenal gland. No subjects had SFCs on the right side only. In patients with fCCM, the presence of SFCs showed a positive correlation with age (P < .001) and number of brain lesions (P < .001). Conclusion Adrenal calcifications identified on CT scans are common in patients with fCCM and may be a clinically silent manifestation of disease.

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Sensitivity of patients with familial cerebral cavernous malformations to therapeutic radiation

Cited by 13 publications

References 4 publications

Synopsis of Guidelines for the Clinical Management of Cerebral Cavernous Malformations: Consensus Recommendations Based on Systematic Literature Review by the Angioma Alliance Scientific Advisory Board Clinical Experts Panel

Synopsis of Guidelines for the Clinical Management of Cerebral Cavernous Malformations: Consensus Recommendations Based on Systematic Literature Review by the Angioma Alliance Scientific Advisory Board Clinical Experts Panel

Trial Readiness in Cavernous Angiomas With Symptomatic Hemorrhage (CASH)

Familial Cerebral Cavernous Malformations Are Associated with Adrenal Calcifications on CT Scans: An Imaging Biomarker for a Hereditary Cerebrovascular Condition

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