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Philippe Gailloud

The Polish neurologist Łucja Frey (1889–1942) is principally remembered for her description of the auriculotemporal nerve (or Frey's) syndrome. She also reported a complex case of spinal cord vascular malformation in 1928, which included one of the earliest accurate anatomopathological depictions of a spinal arteriovenous malformation as well as the first known observation of double synchronous spinal cord vascular malformation.

An abbreviated translation of Frey's report is followed by a discussion of the place occupied by her contribution in the development of our current understanding of spinovascular disorders.

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Monica Smith Pearl, Rafael Tamargo and Philippe Gailloud

The angiographic appearance of an intracranial arterial occlusion is typically distinct from that of a saccular aneurysm, with only a few reported cases of occlusion simulating aneurysm. At the same time, a small percentage of symptomatic intracranial aneurysms present with a stroke. Accurate diagnosis of these conditions is crucial, as their treatment differs. The authors report on a case of middle cerebral artery occlusion that mimicked the appearance of an aneurysm on angiography in the setting of acute stroke. The true diagnosis was not elucidated until repeated angiography 6 months later revealed recanalization of the previously occluded middle cerebral artery branch. This angiographic pitfall is important to consider when acute stroke is suspected as the mode of presentation of a saccular aneurysm.

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Patrik Gabikian, Richard E. Clatterbuck, Philippe Gailloud and Danielle Rigamonti

✓ Blue rubber-bleb nevus syndrome (BRBNS) is a developmental disorder that originally was identified by the presence of distinctive cutaneous and gastrointestinal hemangiomas. More recently it has been recognized that the number of affected organs is larger and that BRBNS includes central nervous system vascular malformations. A 52-year-old woman in whom intracranial vascular malformations had been diagnosed earlier presented for evaluation. At birth, several blue nevi had been noted on her tongue, lips, and neck. Cerebral angiography demonstrated an extensive network of developmental venous anomalies and a left anterior sinus pericranii. The literature on BRBNS and developmental venous anomalies is reviewed.

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Edison P. Valle, Rafael J. Tamargo and Philippe Gailloud

The cases of 2 children with true aneurysmal subarachnoid hemorrhages (SAHs) and initial false-negative angiograms are reported. In both cases, the initial angiogram was of adequate technical quality and included the projections on which aneurysms were later documented. There was no significant vasospasm at the time of initial angiography; therefore, transient aneurysm sac thrombosis was the most likely explanation for the initial false-negative studies. It is particularly interesting to note that 1 of the 2 patients had a pattern of hemorrhage compatible with the most limited definition of a perimesencephalic SAH, that is, a small prepontine cistern hemorrhage. If a second angiogram had been deemed unnecessary based on that criterion alone, a ruptured basilar tip aneurysm would have escaped detection and treatment.

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Lori C. Jordan, George I. Jallo and Philippe Gailloud

✓ The authors report the case of a 4-year-old boy with a spontaneous intracerebral hemorrhage (ICH) related to an arteriovenous malformation (AVM) that was not found with good-quality magnetic resonance (MR) imaging and MR angiography. Both modalities were used serially in the acute phase and at 2 and 7 months of follow-up. Digital subtraction angiography identified the peripheral AVM when the patient experienced rehemorrhaging 1 year after his initial presentation. This case illustrates the need for a complete diagnostic evaluation including conventional angiography in cases of idiopathic ICH in children.

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Ingrid M. Burger, Rafael J. Tamargo, Jennifer Broussard and Philippe Gailloud

✓The authors report on the case of a 28-year-old woman presenting with an intraosseous arteriovenous fistula (AVF) located in the left parietal bone. The fistula was formed by direct arteriovenous shunts connecting branches of the left middle meningeal and superficial temporal arteries with a parietal diploic vein. Drainage occurred through both the external and internal jugular venous systems. Therapy consisted of combined surgical and endovascular approaches. The results of a pathological examination of the resected AVF showed mild enlargement of the diploic space. The angiographic appearance, pathological anatomy, and treatment of this rare lesion are discussed, as is a possible relationship between diploic AVFs and the development of aneurysm bone cysts.

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Anshuman Bansal, Philippe Gailloud, Lori Jordan and Diego San Millán Ruíz

The authors present the case of an infant harboring a vein of Galen arteriovenous malformation with conspicuous cerebral calcifications that progressively regressed after staged endovascular obliteration of the lesion. The role of venous hypertension and hydrocephalus secondary to the arteriovenous shunt are discussed to explain the formation and regression of the cerebral calcifications.

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Gary L. Gallia, Carolyn Moore, Lori Jordan, Philippe Gailloud and George I. Jallo

✓Neonatal intracranial aneurysms are rare. The authors report the case of a 4-week-old girl who presented with left-eye ptosis and proptosis. Computerized tomography scanning and magnetic resonance imaging demonstrated a mass involving the left cavernous sinus and middle cranial fossa. Cerebral angiography revealed a large complex left cavernous carotid artery (CA) aneurysm. The patient underwent endovascular treatment in which detachable coils and n-butyl cyanoacrylate glue were used to achieve complete obliteration of the aneurysm. To the authors' knowledge, this is the first reported neonatal intracranial aneurysm originating from the cavernous CA and treated endovascularly. The authors review the literature on neonatal intracranial aneurysms.

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Richard E. Clatterbuck, Bernard Cohen, Philippe Gailloud, Kieran Murphy and Daniele Rigamonti

✓ Recently, several groups of authors have described mutations in the Krevyl interaction—trapped 1 (KRIT1) gene in families in whom cerebral cavernous malformations (CCMs) are present. In a number of French kindreds harboring familial CCMs, cutaneous as well as cerebral manifestations of this autosomal-dominant disorder were demonstrated. Involvement of other tissues has been poorly described. The authors present the proband, in an affected family with a previously reported KRIT1 mutation, in whom vertebral hemangiomas in addition to cerebral and cutaneous lesions were found. One of the vertebral lesions was associated with a large cutaneous lesion. This combination of vertebral and overlying cutaneous lesions suggests segmental disease expression as the result of a second hit during development, implying loss of function as the relevant molecular pathogenic mechanism. This case illustrates that tissue involvement outside the nervous system must be considered when treating patients with familial CCMs.