Ruptured aneurysm of the anterior spinal artery associated with pseudoxanthoma elasticum

Kito, Kenichi; Kobayashi, Naotoshi; Mori, Nobuhiko; Kohno, Hiroshi

doi:10.3171/jns.1983.58.1.0126

Cited by 52 publications

(16 citation statements)

References 12 publications

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“…It is conceivable that this obstruction of the left internal carotid artery may have increased the strain in the vertebrobasilar system. Other craniocervical vascular malformations described in patients with PXE are fusiform cervical aneurysm [24], pontine arteriovenous malformation [25], bilateral calcified common carotid aneurysms [12], carotid rete mirabile [26], and an aneurysm of the anterior spinal artery [27]. The last patient had stenosis of the abdominal aorta and dilatation of internal thoracic arteries.…”

Section: Discussionmentioning

confidence: 99%

Prevalence of Symptomatic Intracranial Aneurysm and Ischaemic Stroke in Pseudoxanthoma Elasticum

Berg

Hennekam²,

Cruysberg

et al. 2000

Cerebrovasc Dis

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Background: Pseudoxanthoma elasticum (PXE) is an heritable connective tissue disorder with clinical manifestations of the ocular, dermal, and cardiovascular system. The purpose of this study was to investigate the prevalence of symptomatic intracranial aneurysms (IAs) and ischaemic stroke (IS) in PXE. Methods: The records of 100 patients with PXE were retrieved. All patients were contacted and data on complications were collected. The literature was reviewed regarding PXE, ISs, and IAs. Results: No patient with PXE had a symptomatic IA as presenting symptom. One patient presented with an IS. During follow-up of 94 of the 100 patients (mean follow-up 17.1 years, range 1–49 years), none presented a symptomatic IA (3,168 retrospective patient observation years and 1,602 prospective patient observation years). Upper gastrointestinal haemorrhage during follow-up occurred in 17 patients, in 1 patient during aspirin use. One patient has IS as presenting symptom and a recurrence during follow-up, and 7 patients had IS during follow-up. All were caused by small-vessel disease. The relative risk of IS in PXE under 65 years compared with the general population was 3.6 (95% confidence interval 3.3–4.0). Conclusions: On the basis of the currently available data, an association between symptomatic IAs and PXE is unlikely. However, the incidence of IS, due to small-vessel disease, was increased. Antiplatelet therapy in patients with PXE may lead to a high incidence of upper gastrointestinal haemorrhages.

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Section: Discussionmentioning

confidence: 99%

Prevalence of Symptomatic Intracranial Aneurysm and Ischaemic Stroke in Pseudoxanthoma Elasticum

Berg

Hennekam²,

Cruysberg

et al. 2000

Cerebrovasc Dis

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“…Central nervous system involvement accounts for many of the most serious complications of PXE [3, 9 . Subarachnoid hemorrhage (SAH) and intracerebral hem orrhage are frequent causes o f death in PXE patients [10][11][12], Although carotid artery aneurysms are most common [3,[11][12][13], aneurysm form ation may also occur in unustal locations, such as the anterior spinal artery [ 14] or bilaeral extracranial carotid arteries [12], SAH from ruptuu' of an arteriovenous m alform ation has also been reporte I in association with PXE [15], Ischemic cerebrovascular disease in PXE may resu t from a variety of mechanisms, and affected patients ofte i have m ultiple risk factors for stroke [3]. Hypertension h; s been reported to be 3-fold more common than in the geieral population [2], presumably the result of either re duced compliance of the arterial system [3,16] or involve m ent of the renal glomeruli [2], Prem ature coronary artery disease [17,18], calcific cardiac valvular disea.-e [19], and severe occlusive disease o f the aorta [6] may occur, the latter often resulting in asymmetric blood pré sure measurem ents, as found in our second patient.…”

Section: Discussionmentioning

confidence: 99%

Recurrent Ischemic Events and Diffuse White Matter Disease in Patients with Pseudoxanthoma Elasticum

et al. 1994

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Pseudoxanthoma elasticum (PXE) is a rare hereditary disorder of elastic tissue with prominent dermatologic, ocular, and vascular manifestations. Although PXE has been associated with ischemic stroke, the clinical and radiologic features have not been fully characterized. We describe two women with PXE, hypertension, and progressive motor and intellectual deterioration related to multiple strokes and transient ischemic attacks beginning at ages 50 and 56. Magnetic resonance imaging in both patients showed widespread and multiple punctate ischemic vascular lesions with diffuse white matter involvement. Although brain infarction in PXE may result from a variety of mechanisms, our patients show the association of this disorder with severe, predominantly small-vessel occlusive disease.

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“…Isolated spinal artery aneurysms are exceedingly rare, and are thought to be related to dissection of the arterial wall probably associated with underlying states leading to inflammation of the vascular wall 2 , as reported in at least five previous cases (Pseudoxanthoma elasticum 5 , fibromuscular dysplasia 6 , Behcet's disease 7 , systemic candidiasis and rheumatoid arthritis 8 ), interestingly all of them located in the anterior spinal artery.…”

Section: Discussionmentioning

confidence: 97%

Ruptured Isolated Spinal Artery Aneurysms Report of Two Cases and Review of the Literature

et al. 2014

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Isolated spinal artery aneurysms are exceedingly rare vascular lesions thought to be related to dissection of the arterial wall. We describe two cases presenting with spinal subarachnoid haemorrhage that underwent conservative management. In the first patient the radiculomedullary branch involved was feeding the anterior spinal artery at the level of D3 and thus, neither endovascular nor surgical approach was employed. Control angiography was performed at seven days and at three months, demonstrating complete resolution of the lesion. In our second case, neither the anterior spinal artery or the artery of Adamkiewicz could be identified during angiography, thus endovascular management was deemed contraindicated. Magnetic resonance imaging showed a stable lesion in the second patient. No rebleeding or other complications were seen. In comparison to intracranial aneurysms, spinal artery aneurysms tend to display a fusiform appearance and lack a clear neck in relation to the likely dissecting nature of the lesions. Due to the small number of cases reported, the natural history of these lesions is not well known making it difficult to establish the optimal treatment approach. Various management strategies may be supported, including surgical and endovascular treatment, but It would seem that a wait and see approach is also viable, with control angiogram and treatment decisions based on the evolution of the lesion.

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Ruptured aneurysm of the anterior spinal artery associated with pseudoxanthoma elasticum

Abstract: ✓ An aneurysm of the anterior spinal artery associated with pseudoxanthoma elasticum is reported. This aneurysm was demonstrated by selective spinal angiography.

Cited by 52 publications

References 12 publications

Prevalence of Symptomatic Intracranial Aneurysm and Ischaemic Stroke in Pseudoxanthoma Elasticum

Prevalence of Symptomatic Intracranial Aneurysm and Ischaemic Stroke in Pseudoxanthoma Elasticum

Recurrent Ischemic Events and Diffuse White Matter Disease in Patients with Pseudoxanthoma Elasticum

Ruptured Isolated Spinal Artery Aneurysms Report of Two Cases and Review of the Literature

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