123I-IMP-SPECT in a Patient with Cerebral Proliferative Angiopathy: A Case Report

Kimiwada, Tomomi; Hayashi, Toshiaki; Shirane, Reizo; Tominaga, Teiji

doi:10.1016/j.jstrokecerebrovasdis.2013.05.038

Cited by 20 publications

(16 citation statements)

References 14 publications

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“…123 I-IMP-SPECT at resting state shows preserved uptake within the vascular lesion, yet lower uptake in the area adjacent to the lesion suggests cerebral ischemia. 7 DSA is gold standard for diagnosis of CPA due to its realtime dynamic flow evaluation capability. The DSA generally shows a nidus size greater than 6 cm with venous drainage not having ectasia, in most cases.…”

Section: Discussionmentioning

confidence: 99%

Cerebral Proliferative Angiopathy: An Uncommon and Misdiagnosed Entity

Tiwari

Garg

Khera

et al. 2020

Journal of Clinical Interventional Radiology ISVIR

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Cerebral proliferative angiopathy (CPA), previously known as diffuse nidus-type arteriovenous malformation (AVM) is an entity distinct from cerebral AVM, characterized by multiple small arterial feeders, large-size nidus involving entire lobe or hemisphere, and no early draining veins with normal brain parenchyma interspersed between the abnormal vessels. It is usually seen in younger age group and is more common in females. We hereby report a case of diffuse cerebral proliferative angiopathy in a 29-year-old man who presented with intracranial hemorrhage. It is important to recognize this entity to avoid aggressive treatment, thus preventing permanent damage to the normal intermingled brain tissue.

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Section: Discussionmentioning

confidence: 99%

Cerebral Proliferative Angiopathy: An Uncommon and Misdiagnosed Entity

Tiwari

Garg

Khera

et al. 2020

Journal of Clinical Interventional Radiology ISVIR

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“…There have been several case reports on patients with CPA, [1][2][3][4][5][6][7][8][9]11,12,15 and one series of 49 patients with this disorder. 10 In that study of 49 patients (mean age 22 years), 45% presented with seizures, 41% with severe and often disabling headaches, 12% with hemorrhage, and 16% with nonhemorrhagic neurological deficits.…”

Section: Discussionmentioning

confidence: 99%

“…5,9,10,15 In addition, angiographically and clinically progressive features have been reported, indicating the progressive nature of CPA. 10,12 These features are similar to those of moyamoya disease, which is an idiopathic, noninflammatory vasculopathy characterized by progressive stenosis or occlusion of the supraclinoid internal cerebral arteries (ICAs).…”

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confidence: 99%

Encephaloduroarteriosynangiosis for cerebral proliferative angiopathy with cerebral ischemia

Kono¹,

Terada²

2014

JNS

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Cerebral proliferative angiopathy (CPA) is a rare clinical entity. This disorder is characterized by diffuse vascular abnormalities with intermingled normal brain parenchyma, and is differentiated from classic arteriovenous malformations. The management of CPA in patients presenting with nonhemorrhagic neurological deficits due to cerebral ischemia is challenging and controversial. The authors report a case of adult CPA with cerebral ischemia in which neurological deficits were improved after encephaloduroarteriosynangiosis (EDAS).A 28-year-old man presented with epilepsy. Magnetic resonance imaging and angiography showed a diffuse vascular network (CPA) in the right hemisphere. Antiepileptic medications were administered. Four years after the initial onset of epilepsy, the patient's left-hand grip strength gradually decreased over the course of 1 year. The MRI studies showed no infarcts, but technetium-99m-labeled ethyl cysteinate dimer ( 99m Tc-ECD) SPECT studies obtained with acetazolamide challenge demonstrated hypoperfusion and severely impaired cerebrovascular reactivity over the affected hemisphere. This suggested that the patient's neurological deficits were associated with cerebral ischemia. The authors performed EDAS for cerebral ischemia, and the patient's hand grip strength gradually improved after the operation. Follow-up angiography studies obtained 7 months after the operation showed profound neovascularization through the superficial temporal artery and the middle meningeal artery. A SPECT study showed slight improvement of hypoperfusion at the focal region around the right motor area, indicating clinical improvement from the operation. The authors conclude that EDAS may be a treatment option for CPA-related hypoperfusion. (http://thejns.org/doi/abs/10.3171/2014.7.JNS132793) Key WorDs • arteriovenous malformation • cerebral ischemia • cerebral proliferative angiopathy • encephaloduroarteriosynangiosis • epilepsy • vascular disordersAbbreviations used in this paper: AVM = arteriovenous malformation; CPA = cerebral proliferative angiopathy; ECA = external carotid artery; EDAS = encephaloduroarteriosynangiosis; ICA = internal cerebral artery; STA = superficial temporal artery; 99m Tc-ECD = technetium-99m-labeled ethyl cysteinate dimer; VEGF = vascular endothelial growth factor.

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“…Perfusion images may play a role in the diagnosis and follow-up of these patients, to assess the extension of ischemic compromise and the efficacy of treatments (3,20) . Several methods were used across case reports and series, such as perfusion-weighted MRI (PW-MRI) (3,6,15,(20)(21)(22) , 123I-IMP-SPECT (21) , 99mTc-ECD-SPECT (18) , Positron Emission Tomography (PET) (23) , and 11C-METH PET (24) .…”

Section: Diagnosismentioning

confidence: 99%

“…Cerebrovascular reserve (CVR), a surrogate measure of adequate blood supply, can be severely impaired around the lesions (15) . Cerebral blood flow (CBF) is believed to be inaccurately elevated due to increased and abnormal vessel structure, with reports that it is actually reduced on 123I-IMP-SPECT analysis (21) . Other methods have shown similar results in publications by different authors (18,(23)(24) .…”

Section: Diagnosismentioning

confidence: 99%

Cerebral proliferative angiopathy: a review / Angiopatia proliferativa cerebral: uma revisão

Nunes

Santos

Aguiar

et al. 2019

Arq Med Hosp Fac Cienc Med Santa Casa São Paulo

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Introduction: Cerebral Arteriovenous Malformations (AVMs) are vascular lesions defined by arteriovenous shunting flowing through a nidus of coiled and tortuous vascular connections. Cerebral Proliferative Angiopathy (CPA) is a rare type of AVM with pathological, clinical, diagnostic features and treatment differences from a typical AVM. We aimed to summarize important features of CPA already described and identify important gaps of evidence. Methods: Data was selected by a search of PUBMED with the term "cerebral proliferative angiopathy". The articles considered relevant were included in this review. We also screened article references and included them as needed. Discussion: CPA shows a diffuse nidus appearance, presence of transdural supply and stenosis of feeding arteries, without dominant feeders or flow-related aneurysms. There is intermingled brain between the vascular spaces. The usual clinical presentation is seizures, headaches, and progressive neurological deficits. Sound diagnosis can be made based on CT, MRI, Angiography and perfusion techniques, which are essential to treatment choices. Invasive treatment is seldom indicated, in order to preserve the normal brain parenchyma, and clinical control of symptoms, associated with a thorough follow-up, is the usual approach in most reported cases. Conclusion: Still much remains to be learned about CPA. Establishing more precise roles of various radiological assessing methods, how they affect the follow-up of patients, and safety and efficacy profiles of different treatment approaches will provide us the tools to give patients a more solid intervention and clearer follow-up.

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123I-IMP-SPECT in a Patient with Cerebral Proliferative Angiopathy: A Case Report

Cited by 20 publications

References 14 publications

Cerebral Proliferative Angiopathy: An Uncommon and Misdiagnosed Entity

Cerebral Proliferative Angiopathy: An Uncommon and Misdiagnosed Entity

Encephaloduroarteriosynangiosis for cerebral proliferative angiopathy with cerebral ischemia

Cerebral proliferative angiopathy: a review / Angiopatia proliferativa cerebral: uma revisão

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