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PubMed2006

Benign intracranial hypertension associated to blood coagulation derangements.

BACKGROUND: Benign Intracranial Hypertension (BIH) may be caused, at least in part, by intracranial sinus thrombosis. Thrombosis is normally due to derangements in blood coagulation cascade which may predispose to abnormal clotting activation or deficiency in natural inhibitors' control. The aim of the study is to examine the strength of the association between risk factors for thrombosis and BIH. PATIENTS AND METHODS: The incidence of prothrombotic abnormalities among a randomly investigated cohort of 17 patients with BIH, was compared with 51 healthy subjects matched for sex, age, body mass index, height and social background. RESULTS: The number of subjects with protein C deficiency was significantly higher in patients than in controls (3 vs 1, p < .001; Fisher Exact Test). Moderate to high titers of anticardiolipin antibodies (beta2-Glycoprotein type I) were found in 8 out of 17 patients. Increased plasma levels of prothrombin fragment 1+2, fibrinopeptide A (FPA), and PAI-1 were demonstrated in patients group (5.7 +/- 1.15 nM vs 0.45 +/- 0.35 nM; 8.7 +/- 2.5 ng/mL vs 2.2 +/- 1.25 ng/mL; 45.7 +/- 12.5 ng/mL vs 8.5 +/- 6.7 ng/mL, respectively; p < .001; Fisher Exact Test). Gene polymorphisms for factor V Leiden mutation, prothrombin mutation 20210 A/G, MTHFR 677 C/T, PAI-1 4G/5G, ACE I/D were detected in 13 patients. DISCUSSION: In agreement with other authors our data suggest a state of hypercoagulability in BIH associated with gene polymorphisms. Our findings also showed that mutations in cardiovascular genes significantly discriminate subjects with a BIH history. The association between coagulation and gene derangements, usually regarded to as cryptogenic, may suggest a possible pathogenetic mechanism in BIH. So, a prothrombotic tendency may exist that would, at least in part, explain some cases of BIH. Although based on a small population, these findings raise the exciting possibility of using these haemostatic factors as markers for selecting high-risk subjects in BIH disease.

Journal Article↗
PubMed1977

Coxsackie A9 focal encephalitis associated with acute infantile hemiplegia and porencephaly.

The factors underlying acute infantile hemiplegia are seldom identified. Coxsackie A9 focal encephalitis was documented for the first time in a 3-month-old infant with fever, hemiconvulsions, and hemiplegia followed by a static motor deficit and epilepsy. It has been suggested that the acute infantile hemiplegia associated with encephalitis results from an arteritis or venous sinus thrombosis with subsequent cerebral infarction. However, this was not observed in our patient. Rather, a series of brain scans, computerized tomograms, and a cerebral angiogram clearly documented the evolution of a focal necrotizing encephaloclastic process resulting in a porencephalic cyst. Serial cerebrospinal fluid viral cultures were necessary to isolate the etiologic agent (tcoxsackie A9). The infant did not have a neutralizing antibody response to the infecting viral agent despite an apparently intact immune system, which possibly may be explained by the developed of immune tolerance or an insufficient amount of infecting viral antigen. This emphasizes that serologic studies alone may not be adequate to document an acute central nervous system viral infection. This patient also typifies the poor prognosis in infants presenting with acute hemiplegia, fever, and convulsions in the absence of cerebrovascular occlusion.

Acute Disease↗
PubMed1986

Neurologic complications of carcinoid.

We reviewed the records of all patients treated for carcinoid tumors at Memorial Sloan-Kettering Cancer Center from 1974 through 1984. Of 219 patients, 90 developed metastatic complications, and of these, 36 developed neurologic complications. Metastases, the most common neurologic complication, included epidural spinal cord compression (14 patients), intracranial metastases (13 patients), leptomeningeal metastases (1 patient), and peripheral nerve lesions (5 patients). Nonmetastatic complications were hepatic encephalopathy (six patients), herpes zoster infection (two patients), cerebral infarction due to septic emboli (one patient), superior sagittal sinus thrombosis (one patient), and carcinoid myopathy (one patient). The carcinoid syndrome was seen in eight patients (4%). Response of neurologic metastases to conventional radiation therapy was usually favorable. We conclude that (1) the frequency and type of neurologic complications associated with carcinoid tumors are similar to those seen with other systemic cancers; (2) CNS metastases are relatively common in patients with metastatic carcinoid (29%); and (3) the carcinoid syndrome is less common than CNS metastasis.

Adolescent↗
PubMed1994

Visual loss from idiopathic intracranial pachymeningitis.

We report three patients with chronic headaches and optic neuropathy due to widespread meningeal thickening shown on enhanced MRI; all had biopsy-proven intracranial pachymeningitis (fibrosclerosis of the meninges). Two patients had bilateral optic neuropathy, elevated CSF protein, and polyclonal serum hypergammaglobulinemia. They developed temporal lobe cortical necrosis or sagittal sinus thrombosis, presumably due to compromised dural venous drainage from extensive meningeal fibrosis. The other patient had multiple cranial nerve palsies and unilateral optic neuropathy with normal CSF. Corticosteroid therapy improved visual function in all three patients, although all had persisting visual deficits. Gadolinium-enhanced MRI was essential in identifying meningeal inflammation and locating suitable biopsy sites.

Adult↗
PubMed1988

Dural arteriovenous malformations and papilledema.

We examined and managed two patients with posterior fossa dural arteriovenous malformations (DAVMs) and papilledema. Both DAVMs had venous drainage into the transverse, straight, and sigmoid dural venous sinuses. The mechanism of papilledema in the first case was presumed venous hypertension resulting in impaired cerebrospinal fluid absorption, as the malformation drained into the single transverse sinus. This was cured by selective arterial embolization of the causative DAVM. The second patient had venous sinus thrombosis that impaired venous drainage despite embolization. A lumboperitoneal shunt was necessary to treat the elevated intracranial pressure.

Cerebrospinal Fluid Shunts↗
PubMed1976

Symposium: contraindications to tympanoplasty. II. An exercise in clinical judgment.

The two primary symptoms of chronic ear disease are otorrhea and hearing loss. The goal of any tympanoplasty is to eradicate disease and improve hearing. These are the primary indications for this procedure. The success or failure of the surgery is influenced by many factors such as the age and general health of the patient, the extent and type of disease present, and whether the upper airway is functioning properly. Contraindications to tympanoplasty will vary from one surgeon to another depending upon his training, philosophy, experience, and surgical skill. It is helpful to think of these contraindications as being either absolute or relative. Absolute contraindications consist of: 1. uncontrolled cholesteatoma; 2. malignant tumors; 3. unusual infections and/or extenuating circumstances; and 4. complications of chronic ear disease, such as meningitis, brain abscess, or lateral sinus thrombosis. Relative contraindications are less well defined: 1. eustachian tube insufficiency or non-function; 2. the uncooperative patient; 3. the dead ear; 4. the better hearing or only hearing ear; 5. the elderly patient; 6. the young child; and 7. the repeated failure case. The otologic surgeon must exercise good clinical judgment in selecting patients for tympanoplasty. This paper deals with some of the more common problems in decision making.

Age Factors↗
PubMed2004

Antithrombotic and thrombolytic therapy for ischemic stroke: the Seventh ACCP Conference on Antithrombotic and Thrombolytic Therapy.

This chapter about treatment and prevention of stroke is part of the 7th ACCP Conference on Antithrombotic and Thrombolytic Therapy: Evidence Based Guidelines. Grade 1 recommendations are strong and indicate that the benefits do, or do not, outweigh risks, burden, and costs. Grade 2 suggests that individual patients' values may lead to different choices (for a full understanding of the grading see Guyatt et al). Among the key recommendations in this chapter are the following: For patients with acute ischemic stroke (AIS), we recommend administration of i.v. tissue plasminogen activator (tPA), if treatment is initiated within 3 h of clearly defined symptom onset (Grade 1A). For patients with extensive and clearly identifiable hypodensity on CT, we recommend against thrombolytic therapy (Grade 1B). For unselected patients with AIS of > 3 h but < 6 h, we suggest clinicians not use i.v. tPA (Grade 2A). For patients with AIS, we recommend against streptokinase (Grade 1A) and suggest clinicians not use full-dose anticoagulation with i.v. or subcutaneous heparins or heparinoids (Grade 2B). For patients with AIS who are not receiving thrombolysis, we recommend early aspirin therapy, 160 to 325 mg qd (Grade 1A). For AIS patients with restricted mobility, we recommend prophylactic low-dose subcutaneous heparin or low molecular weight heparins or heparinoids (Grade 1A); and for patients who have contraindications to anticoagulants, we recommend use of intermittent pneumatic compression devices or elastic stockings (Grade 1C). In patients with acute intracerebral hematoma, we recommend the initial use of intermittent pneumatic compression (Grade 1C+). In patients with noncardioembolic stroke or transient ischemic attack (TIA) [ie, atherothrombotic, lacunar or cryptogenic], we recommend treatment with an antiplatelet agent (Grade 1A) including aspirin, 50 to 325 mg qd; the combination of aspirin and extended-release dipyridamole, 25 mg/200 mg bid; or clopidogrel, 75 mg qd. In these patients, we suggest use of the combination of aspirin and extended-release dipyridamole, 25/200 mg bid, over aspirin (Grade 2A) and clopidogrel over aspirin (Grade 2B). For patients who are allergic to aspirin, we recommend clopidogrel (Grade 1C+). In patients with atrial fibrillation and a recent stroke or TIA, we recommend long-term oral anticoagulation (target international normalized ratio, 2.5; range, 2.0 to 3.0) [Grade 1A]. In patients with venous sinus thrombosis, we recommend unfractionated heparin (Grade 1B) or low molecular weight heparin (Grade 1B) over no anticoagulant therapy during the acute phase.

Cerebral Infarction↗
PubMed2004

Pseudotumor cerebri in children with sickle cell disease: a case series.

Headache is a frequent symptom in sickle cell disease (SCD) that usually is attributable to anemia or cerebrovascular disease. We report 3 pediatric patients with SCD (1 patient with SCD-SC and 2 patients with SCD-SS) who presented with headache and were diagnosed with pseudotumor cerebri (PC). All 3 patients had elevated opening pressures during a lumbar puncture with normal cerebrospinal fluid studies. Magnetic resonance imaging revealed no evidence of hydrocephalus or arteriopathy in all 3 cases. Magnetic resonance venograms performed in 2 of the patients at diagnosis revealed no evidence of cerebral sinus thrombosis. Each patient received a thorough ophthalmologic examination. A diagnostic funduscopic examination revealed bilateral papilledema without signs of retinopathy in all 3 patients. There were no clinically significant changes in visual acuity or abnormalities of color vision in any patient. Goldmann or Humphrey visual-field assessment was abnormal only in patient 1, who demonstrated bilaterally enlarged blind spots at diagnosis and later developed reduced sensitivity in the inferomedial quadrant of the left eye in an arcuate pattern (which later resolved). The diagnosis of PC was made in all 3 patients, and acetazolamide treatment was started. Two of the patients' symptoms resolved completely with medical treatment, whereas the third patient's symptoms improved. None of these patients had permanent visual-field deficits as a result of their syndrome. PC has been reported in several other types of anemia including SCD-SC, but these cases are the first reported in conjunction with pediatric SCD. Early recognition of the signs and symptoms of PC in patients with SCD who present with headache can expedite proper diagnosis and treatment and prevent long-term ophthalmologic sequelae.

Adolescent↗
PubMed1994

Thrombolytic therapy in acute stroke.

PURPOSE: To report the safety and efficacy of local, direct, intra-arterial and intravenous fibrinolysis treatment in selected cases of clinically symptomatic patients with acute occlusion of the intracranial cerebral arteries and dural sinuses. METHODS: Patients with acute progressive neurological deterioration, in spite of systemic anticoagulation and/or antiplatelet medications, presenting with occlusion of a major intracranial cerebral artery or dural sinus were tested. From a transfemoral approach through a guiding catheter, a 2.5F microcatheter was guided directly into the intracranial cerebral circulation and embedded within the clot. Infusion of urokinase was then performed directly into the thrombus until lysis was attained. RESULTS: In 36 total patients, 27 cases were treated for an acute arterial occlusion in 45 vascular territories. Clinically, there was neurological improvement in 18 (66.7%) cases. Complications directly related to therapy included symptomatic intracranial hemorrhage in three cases (11.1%), which included 1 case (3.7%) of vessel perforation. In 8 (29.6%) patients, there was no evidence of clinical improvement, and in long-term follow-up there were 9 (33.3%) patient deaths. Nine patients were treated for an intracerebral dural sinus thrombosis in ten vascular territories by local urokinase infusion. In 7 (77.8%) cases, there was angiographic evidence of clot lysis and clinical improvement of the patient's neurological condition. Minor complications including infection and noncerebral sites of bleeding occurred in 3 (33.3%) patients, requiring adjustment in urokinase infusion therapy. CONCLUSION: Local, direct intra-arterial or intravenous infusion of thrombolytic drugs for treatment of stroke patients may improve overall patient morbidity and mortality related to acute thromboembolic disease in the central nervous system. Further clinical studies are warranted to evaluate this form of therapy.

Acute Disease↗
PubMed1990

Treatment of intraventricular hemorrhage using urokinase.

Four patients with severe intraventricular hemorrhage (IVH) were treated using urokinase administered into the lateral ventricles via a ventricular drainage catheter. All patients were female and of ages ranging from 24 to 53 years. The primary diseases were hemorrhagic infarction, moyamoya disease, sinus thrombosis, and thalamic bleeding. Urokinase administration was initiated at 1.3 days average after occurrence of IVH and continued for 3.3 days average in doses of 12,000-96,000 IU per day. Average clot lysis times from IVH, as assessed by computed tomography, were 5.0 +/- 0.8, 5.0 +/- 1.4, and 6.0 +/- 0.8 days for the fourth, the third, and the lateral ventricles, respectively. All patients suffered from meningitis which was probably caused by urokinase administration through a ventricular catheter. However, this was successfully treated by changing the antibiotics. There was no general bleeding tendency or intraventricular rebleeding due to urokinase administration, and none of the ventricular catheters were obstructed by clots throughout the course. The final outcome was good recovery in two patients, severe disability in one, and persistent vegetative state in one. These results correlated well with the consciousness level seen before ventricular drainage in each patient. Consequently, we are convinced that urokinase administration can prevent the harmful effects of IVH and that urokinase is useful not only for lysing ventricular clots but also for maintaining the patency of the ventricular catheter, which is important for control of intracranial pressure in the acute stage of severe IVH.

Adult↗
PubMed1990

[Dural arteriovenous malformation in the posterior fossa presenting with multiple intracerebral hematomas. Case report].

A 59-year-old female was hospitalized because of disturbance of consciousness and convulsive seizures. She had taken a hormonal drug for 15 months after breast cancer surgery. A computed tomography scan revealed multiple high-density areas in the left temporal and frontal and the right parietal lobes. Angiography showed a dural arteriovenous malformation (AVM) in the posterior fossa fed by the occipital and the middle meningeal arteries and draining into the transverse sinus. It also demonstrated occlusion of the left sigmoid sinus in the venous phase. She complained of headache in the occipital region and dizziness. On day 13, the left occipital artery was ligated and cut, and then abnormal arterial anastomoses around the lesion were coagulated. After surgery, clinical symptoms disappeared. The etiology of dural AVM is controversial, but in this case it is suspected that sinus thrombosis due to the drug caused the dural AVM. The authors discuss the etiology and treatment of dural AVM in the posterior fossa.

Arteriovenous Malformations↗
PubMed1998

Sigmoid sinus dural arteriovenous malformation resulting from jugular foramen schwannoma--case report.

A 69-year-old male presented with a jugular foramen schwannoma occluding the sigmoid sinus and associated with sigmoid sinus dural arteriovenous malformation. The patient presented with dizziness and pulsatile tinnitus following an extended period of hearing loss beginning several years before. Both lesions were resected successfully after transarterial embolization of the malformation. The sequence of symptom development suggests the dural sinus thrombosis caused the dural arteriovenous malformation.

Aged↗
PubMed2000

Hypoglossal neurinoma--two case reports.

Two patients presented with hypoglossal neurinoma extending both intra- and extracranially. A 63-year-old male presented with right trigeminal neuralgia and hypoglossal nerve paresis. The intracranial part of the tumor was removed totally via a suboccipital craniectomy. Over-coagulation of the venous collaterals, particularly the emissary veins, resulted in dural venous sinus thrombosis and cerebellar infarction. Unfortunately this patient died. A 48-year-old male presented with pareses of the VII, IX, X, XI, and XII cranial nerves and cerebellar sign. The tumor extended both extra- and intracranially, and was completely removed by opening the hypoglossal canal and the jugular foramen without over-coagulation of the venous collaterals. Preservation of the venous collaterals is very important for the prevention of postoperative venous complications.

Cerebral Angiography↗
PubMed1992

MR angiography of the head and neck: value of two-dimensional phase-contrast projection technique.

MR angiography is commonly performed by using two- and three-dimensional time-of-flight and three-dimensional phase-contrast techniques. These procedures require long examination times and processing of imaging data by computing maximum intensity projections. Two-dimensional phase-contrast projection angiography has neither of these disadvantages. We analyzed the value of this technique for head and neck MR angiography in 84 patients and 15 control subjects. Patients were examined to resolve specific clinical questions such as the presence of arteriovenous malformations (20 cases), patency of carotid and vertebral arteries (35 cases), patency of the superior sagittal sinus (14 cases), patency of saphenous vein bypass grafts (11 cases), and vascularity of masses (four cases). Conventional angiograms were available for correlation in 22 patients. Two-dimensional phase-contrast projection angiograms were generated by using a gradient-recalled-echo sequence sensitized to flow with the use of flow-encoding gradients. Projection MR angiograms were obtained in approximately 3.5 min by combining images obtained with flow-encoding gradients applied along the axes defining the image plane. MR angiograms were subjectively evaluated by three observers without reference to routine MR images or conventional angiograms. High-quality studies were obtained in 93% of control subjects and 90% of patients examined. The findings based on phase-contrast angiography were confirmed with conventional angiography in 21 of 22 patients for whom conventional angiograms were available. Evaluation of vascular grafts and of the patency of major cranial vessels was easily done with two-dimensional phase-contrast angiography and was useful in postoperative follow-up examinations. We also advocate its use for superior sagittal sinus thrombosis and follow-up examinations in patients with arteriovenous malformations.

Arteriovenous Malformations↗
PubMed1992

MR angiography in children with cerebral neurovascular diseases: findings in 31 cases.

OBJECTIVE: We evaluated the suitability of MR angiography for routine use in children with suspected intracranial vascular disease. SUBJECTS AND METHODS: Thirty-one children, 6 months to 14 years old, with intracranial lesions or clinically suspected vascular malformations were studied prospectively with conventional MR imaging and time-of-flight MR angiography. In nine cases, MR angiographic findings were verified with digital subtraction angiography or conventional angiography. All MR studies were performed on a 1.5-T MR system using a circularly polarized head coil. RESULTS: Arterial MR angiography, performed in 24 cases, revealed congenital abnormalities of the arterial vessels in 20 cases. Vessel stenosis was observed in nine patients, and displacement of intracranial arteries due to tumors could be seen in 10 patients. Seven children had no abnormal findings. Venous MR angiography was performed in seven children, with depiction of sinus thrombosis in six cases. The comparative analysis of MR angiography and digital subtraction angiography showed equivalent results in nine patients; in one patient the degree of stenosis was overestimated with MR angiography. CONCLUSION: MR angiography, when combined with MR imaging, reveals information about soft-tissue and vascular structures in a single setting. At this point, MR angiography can replace invasive conventional angiography or digital subtraction angiography only in selected cases because of software and hardware limitations. Arterial or venous MR angiography can be helpful as an additional scan in MR examinations of children with suspected cerebral neurovascular diseases, and its noninvasive nature makes it well suited for routine use in children.

Adolescent↗
PubMedDate not supplied

[Behçet's disease in childhood].

OBJECTIVE: To make Pediatricians aware of the fact that they must investigate Behçet's disease while performing differential diagnosis of recurrent aphthous stomatitis, even though it is a vasculitis of rare occurrence in early life. METHODS: Between June 1996 and December 2000, we retrospectively evaluated seven patients of our Pediatric Rheumatology Unit. Demographic, clinical, and laboratory data as well as data regarding treatment and follow-up were presented. RESULTS: Five out of seven patients were female (71.4%), four were non-Caucasian (57.1%), the mean age at onset was 8 years and 11 months (variation of 6 months to 13 years and 8 months), the mean period until diagnosis was 2 years and 3 months (variation of 2 months to 8 years) and the mean follow-up period was 4 years and 2 months (three patients without follow-up). The major criteria of diagnosis were: oral ulcers in seven patients (100%), genital ulcers in three patients (42.8%), ophthalmic alterations in four patients (57.1%) cutaneous vasculitis in one patient (14.2%) and positive pathology test in one patient (14.2%). The minor criteria were: arthralgia/arthritis in five patients (71.4%), family history in two patients (28.5%), and sagittal sinus thrombosis in one patient (14.2%). The initial symptoms included recurrent aphthous stomatitis (more than three painful aphthous ulcers episodes in the period of 1 year), genital ulcers, arthralgia, fever and weight loss. The laboratory findings were: mild anemia in 1/6 patients, ESR>25 in 3/6 patients, increased serum gammaglobulin level in 2/4 patients, B5 histocompatibility antigen in 2/7 patients. The treatment included corticosteroids for 5/7 patients (4 oral, 1 intravenous and one local use), thalidomide for 4/7 patients, colchicine for 2/7 patients and dapsone for 1/7 patient. The outcome was favorable in 4/6, and 3/6 patients presented relapse. CONCLUSION: Our results confirm the importance of considering the diagnosis of Behçet's disease in patients with recurrent oral and genital ulcers.

English Abstract↗
PubMed1994

Intracranial hypertension in a dieting patient.

We report a case of encephalopathy with paranoid psychosis in association with intracranial hypertension. This occurred in a patient whose diet consisted almost solely of walnuts, ginseng tea, and vitamin A supplements. The patient was found to be severely iron- and vitamin B12-deficient. She was vitamin A toxic. Venous sinus thrombosis was also present. Symptoms remitted with serial lumbar punctures, normalization of diet, and repletion of vitamin B12 and iron stores. Physicians should be alerted to the possibility of a potentially confusing clinical presentation with coexistent and seemingly mutually exclusive neurologic conditions in patients with extremely restricted or fad diets.

Adult↗