Discrepancy Between [18F]fluorodeoxyglucose and 11C-methionine Positron Emission Tomography Findings in Sturge-Weber Syndrome -Case Report-

Ichinose, Tsutomu; Tsuyuguchi, Naohiro; Morino, Michiharu; Sunada, Ichiro; Ohata, Kenji; Takami, Toshihiro; Shimonishi, Yoshihiro; Kawabe, Joji; Shiomi, Susumu; Hara, Mitsuhiro

doi:10.2176/nmc.43.461

Cited by 10 publications

(4 citation statements)

References 12 publications

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“…In our study, the increased LEU uptake in the angioma region was mainly driven by the increase in the VD of the tracer, suggesting increased amino acid transport. The marked increase of LEU VD may be due to the retention of the amino acid by increased vascular beds, disruption of the BBB, increased capillary permeability, and/or increased expression of the large amino acid transporter (LAT1) 23,25 . Contrast enhancement of the leptomeningeal angioma on MRI suggests BBB disruption, which itself can lead to increased amino‐acid uptake 26 .…”

Section: Discussionmentioning

confidence: 99%

“…In contrast, FDG uptake is not sensitive to BBB disruption, 29,30 thus, glucose metabolic rates on PET may remain low even in the case of impaired BBB. Accumulation of 11 C‐methionine (which, similar to leucine, is a substrate of LAT1) on PET was reported in the angioma region of an adult with SWS, 25 but quantification of methionine uptake has not been performed. The mechanism of the increase in the K‐complex macroparameter also remains to be determined.…”

Section: Discussionmentioning

confidence: 99%

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Increased L‐[1–¹¹C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study

Alkonyi

Chugani

Muzik

et al. 2011

Journal of Neuroimaging

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Background and Purpose We used L-[1-11C]leucine (LEU) positron emission tomography (PET) to measure amino acid uptake in children with Sturge-Weber syndrome (SWS), and to relate amino acid uptake measures with glucose metabolism. Methods LEU and 2-deoxy-2[18F]fluoro-D-glucose (FDG) PET were performed in 7 children (age: 5 months-13 years) with unilateral SWS. Asymmetries of LEU uptake in the posterior brain region, underlying the angioma, and in frontal cortex, were measured and correlated with glucose hypometabolism. Kinetic analysis of LEU uptake was performed in 4 patients. Results Increased LEU standard uptake value (SUV, mean: 15.1 %) was found in the angioma region in 6 patients, and smaller increases in LEU SUV (11.5 %) were seen in frontal cortex in four of the 6 patients, despite normal glucose metabolism in frontal regions. High LEU SUV was due to both increased tracer transport (3/4 patients) and high protein synthesis rates (2/4). FDG SUV asymmetries in the angioma region were inversely related to LEU SUV asymmetries (r=−0.83, p=0.042). Conclusions Increased amino acid uptake in the angioma region and also in less affected frontal regions may provide a marker of pathological mechanisms contributing to chronic brain damage in children with SWS.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Increased L‐[1–¹¹C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study

Alkonyi

Chugani

Muzik

et al. 2011

Journal of Neuroimaging

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“…Brain positron emission tomography (PET) can follow the extent of cortical dysfunction [66]. Furthermore, single-photon emission computed tomography (SPECT) can be used to detect cerebral blood flow asymmetry which cooperates in being responsible for epileptogenic attack [58]. Both PET and SPECT have been largely used for monitoring and prognostic purposes, being able to detect a deterioration of the glucose metabolism or a decline in the brain perfusion, respectively, in the case of disease progression (namely, epilepsy) as well as an improvement of these parameters in the case of recovery from seizures Fig.…”

Section: Diagnostic Toolsmentioning

confidence: 99%

Sturge-Weber syndrome: an update on the relevant issues for neurosurgeons

et al. 2020

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Purpose Sturge-Weber syndrome (SWS) is a neurocutaneous facomatosis characterized by facial and leptomeningeal angioma, glaucoma, seizures, and neurological disability. Therefore, a challenging multidisciplinary interaction is required for its management. The goal of this paper is to review the main aspects of SWS and to present an illustrative pediatric series. Methods The pertinent literature has been analyzed, focused mainly on etiopathogenesis, pathology, clinical features, diagnostic tools, management, and outcome of the disease. Moreover, a series of 11 children operated on for refractory epilepsy between 2005 and 2015 (minimum follow-up 5 years, mean follow-up 9.6 years) is reported. The series consists of six boys and five girls with 6.5-month and 16.2-month mean age at seizure onset and at surgery, respectively. Seizures affected all children, followed by hemiparesis and psychomotor delay (81%), glaucoma (54%), and other neurological deficits (45%). Results All children underwent hemispherectomy (anatomical in three cases, functional in two cases, hemispherotomy in six cases); one patient needed a redo hemispherotomy. Mortality was nil; disseminated intravascular coagulation and interstitial pneumonia occurred in one patient each; three children had subdural fluid collection. Eight patients (72%) are in the ILAE Class 1 (completely seizure and aura free), two in Class 2 (only auras, no seizure), and one in Class 3 (1-3 seizure days per year). AEDs discontinuation was possible in 73% of cases. The most important news from the literature concerned the pathogenesis (role of the mutation of the GNAQ gene in the abnormal SWS vasculogenesis), the clinical findings (the features and pathogenesis of the stroke-like episodes are being understood), the diagnostic tools (quantitative MRI and EEG), and both the medical (migraine, seizures) and surgical management (epilepsy). The epileptic outcome of SWS patients is very good (80% are seizure-free), if compared with other hemispheric syndromes. The quality of life is affected by the neurological and cognitive deficits. Conclusions SWS still is an etiological and clinical challenge. However, the improvements over the time are consistent. In particular, the neurosurgical treatment of refractory epilepsy provides very good results as long as the indication to treatment is correct.

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“…19 Another PET tracer, 11C-methionine (MET) has useful positive and negative findings in lesion-related epilepsy. Conversely to the high MET uptake over the brain region with focal cortical dysplasia 24 and angiomas (eg., Sturge-Weber syndrome), 15 patients with temporal lobe epilepsy associated with dysembryoplastic neuroepithelial tumors (DNETs) presents negative MET uptake over the lesion, which helps to differentiate the tumor from other lesions. 21 In the future, new tracers with more specificity to the EZ should be developed, but this perspective depends on enhancement in the comprehension of epilepsy patophysiological phenomena such as neurovascular coupling, metabolism and neuroreceptors.…”

Section: Positron Emission Tomography (Pet)mentioning

confidence: 99%

Neurovascular coupling and functional neuroimaging in epilepsy

Junior

Wichert‐Ana

Silva

et al. 2009

J. epilepsy clin. neurophysiol.

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Introduction:The neural regulation of the microcirculation is done by the functional neurovascular unit that is composed of vascular, astroglial and neuronal cells. The neurovascular unit represents the interface between the Central Nervous System and the Vascular System. Objective: This paper reviews the literature on functional neuroimaging with a particular focus on the mechanisms of the neurovascular coupling. Conclusions: Functional neuroimaging techniques as functional MRI, SPECT and PET distinguish metabolic and physiological processes underlying normal and abnormal events, based on neurovascular coupling. Although these techniques still have limitations in temporal and spatial resolution, they have considerably reduced the need for intracranial electrodes or invasive functional tests in the presurgical evaluation for intractable epilepsy. Recently, new techniques as optical approaches (measurement of intrinsic optical signals and near infrared spectroscopy) have increased both temporal and spatial resolutions. The use of such techniques in animal models has yielded experimental evidence for a neurovascular coupling in normal and epileptic conditions.

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Discrepancy Between [18F]fluorodeoxyglucose and 11C-methionine Positron Emission Tomography Findings in Sturge-Weber Syndrome -Case Report-

Cited by 10 publications

References 12 publications

Increased L‐[1–¹¹C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study

Increased L‐[1–¹¹C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study

Sturge-Weber syndrome: an update on the relevant issues for neurosurgeons

Neurovascular coupling and functional neuroimaging in epilepsy

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Discrepancy Between [18F]fluorodeoxyglucose and 11C-methionine Positron Emission Tomography Findings in Sturge-Weber Syndrome -Case Report-

Cited by 10 publications

References 12 publications

Increased L‐[1–11C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study

Increased L‐[1–11C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study

Sturge-Weber syndrome: an update on the relevant issues for neurosurgeons

Neurovascular coupling and functional neuroimaging in epilepsy

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Product

Resources

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Increased L‐[1–¹¹C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study

Increased L‐[1–¹¹C] Leucine Uptake in the Leptomeningeal Angioma of Sturge‐Weber Syndrome: A PET Study