ABSTRACT
OBJECTIVES: Superficial temporal artery (STA)-middle cerebral artery (MCA) bypass operation is an effective treatment for patients with Moyamoya disease, and the hemodynamic parameters are reported to be improved after operation. However, there is no report concerning hemodynamic changes from the viewpoint of the preoperative anatomical structure of grafts. In this study, we evaluated the correlation between the preoperatively estimated blood flow of the graft obtained through image-based computational fluid dynamics (CFD) analysis and the hemodynamic changes in the acute phase after revascularization. MATERIALS AND METHODS: A total of 30 hemispheric sides of 23 patients were examined. The blood flow, that is, flow rate (FR) of the STA branches that were anastomosed to the MCA was evaluated using CFD analysis based on computed tomography (CT) angiography imaging data. The correlations between the FR and the hemodynamic changes in the acute phase after revascularization obtained through CT perfusion were assessed. RESULTS: The preoperatively estimated FR of the graft was moderately correlated with the changes in the mean transit time significantly and weakly correlated with those in the cerebral blood flow and cerebral blood volume. In addition, the FR was strongly correlated with age and the diameter of the STA from the origin to the bifurcation. CONCLUSION: The preoperatively estimated FR of the graft obtained through image-based CFD analysis contributed to the improvement of the mean transit time after revascularization. Because the FR of the graft was associated with the diameter of the STA, the size of the STA might be an important factor in postoperative hemodynamic changes. This might lead to the risk assessment of acute drastic hemodynamic changes as cerebral hyperperfusion, and consequently, better surgical outcomes might be expected.
Subject(s)
Cerebral Revascularization , Cerebrovascular Circulation , Hemodynamics , Middle Cerebral Artery/surgery , Moyamoya Disease/surgery , Temporal Arteries/surgery , Adolescent , Adult , Blood Flow Velocity , Cerebral Angiography , Cerebral Revascularization/adverse effects , Child , Child, Preschool , Computed Tomography Angiography , Female , Humans , Hydrodynamics , Male , Middle Aged , Middle Cerebral Artery/diagnostic imaging , Middle Cerebral Artery/physiopathology , Models, Cardiovascular , Moyamoya Disease/diagnostic imaging , Moyamoya Disease/physiopathology , Patient-Specific Modeling , Perfusion Imaging , Predictive Value of Tests , Retrospective Studies , Temporal Arteries/diagnostic imaging , Temporal Arteries/physiopathology , Treatment Outcome , Young AdultABSTRACT
The COL2A1 gene encodes the alpha-1 chain of procollagen type 2. Pathogenic variants in the COL2A1 gene are associated with several different types of skeletal dysplasia collectively known as type 2 collagenopathies. Type 2 collagenopathies have an autosomal dominant inheritance. Some germline or somatogonadal mosaicism cases have been reported. We investigated whether somatogonadal mosaicism occurred in a family with two children suspected of type 2 collagenopathies or related diseases. First, we detected a pathogenic variant in the COL2A1 gene in the two affected children by whole exome sequencing (WES). Next, we performed targeted deep sequencing to their parents without the variant by WES. A low level of COL2A1 mosaicism was revealed in the mother's tissues. We concluded that the mother had somatogonadal mosaicism with the COL2A1 mutation arose in the epiblast, and that the intrafamilial recurrence rate of the disease by the somatogonadal mosaicism was higher than by the germline mosaicism. This report suggests that parental low-level mosaicism should be evaluated in those parents with children carrying de novo germline mutations and the targeted deep sequencing is useful to detect them.
Subject(s)
Collagen Type II/genetics , Exome Sequencing , Mosaicism , Osteochondrodysplasias/genetics , Female , Genes, Dominant/genetics , Germ-Line Mutation/genetics , High-Throughput Nucleotide Sequencing/methods , Humans , Male , Osteochondrodysplasias/diagnosis , Osteochondrodysplasias/pathology , Parents , RecurrenceABSTRACT
OBJECTIVES: Arterial spin labeling (ASL) is a magnetic resonance imaging (MRI) technique used to assess cerebral perfusion. When tissue perfusion is impaired, such as in Moyamoya disease, a hyperintense band called the arterial transit artifact (ATA) may occur, which interferes with accurate measurements on ASL-MRI. In this study, we evaluated the correlation of ATAs with magnetic resonance angiography (MRA) and single-photon emission computed tomography (SPECT) imaging results in Moyamoya disease. The aim of our study was to elucidate the pathophysiology of ATAs and risk factors for high ATA scores. MATERIALS AND METHODS: This retrospective study included 28 patients (56 hemispheres) with Moyamoya disease treated at our institution. MRI, MRA, ASL perfusion, and N-isopropyl-[123I] b-iodoamphetamine (123I-IMP) SPECT were performed. In order to semi-quantitatively evaluate the degree of ATA, the ATA scores were measured according to the number of hyperintense signal bands in the cerebral cortex. The relationship between the ATA scores and clinical and radiological factors were analyzed. RESULTS: Regional cerebral blood flow (rCBF) determined with ASL weakly correlated with that determined by 123I-IMP SPECT (ρ=0.31, p=0.027). There was no significant association between the ATA scores and rCBF values determined with 123I-IMP SPECT (p=0.872, 0.745, 0.743 at PLD1000 (post-labeling delay), 1500, and 2000, respectively). However, there was a significant correlation between ATA scores and MRA scores (ρ=0.427 p=0.001; ρ=0.612 p=0.001; ρ=0.563 p=0.001 at PLD1000, 1500, and 2000, respectively). An analysis of patient background characteristics revealed a significantly higher incidence of high ATA scores in female patients, patients with high MRA scores, and patients with a distinguishable ivy sign. A multivariate analysis confirmed that female sex, high MRA score, and presence of an ivy sign were risk factors for high ATA scores. CONCLUSION: ATA scores were moderately correlated with MRA scores, and presence of an ivy sign was the most predictive factor for high ATA scores. A high ATA score determined using ASL in a patient with Moyamoya disease might suggest an advanced disease stage and a reduction in cerebrovascular reserve capacity.
Subject(s)
Artifacts , Cerebral Angiography/methods , Cerebral Arteries/diagnostic imaging , Cerebrovascular Circulation , Magnetic Resonance Angiography , Moyamoya Disease/diagnostic imaging , Myocardial Perfusion Imaging/methods , Spin Labels , Tomography, Emission-Computed, Single-Photon , Adult , Blood Flow Velocity , Cerebral Arteries/physiopathology , Female , Humans , Iofetamine/administration & dosage , Male , Middle Aged , Moyamoya Disease/physiopathology , Predictive Value of Tests , Radiopharmaceuticals/administration & dosage , Reproducibility of Results , Retrospective StudiesABSTRACT
OBJECTIVES: Balloon test occlusion (BTO) is performed to evaluate ischemic tolerance for large and giant cerebral aneurysms and head and neck tumors that may require parent artery occlusion. However, ischemic tolerance for the temporary test occlusion does not always guarantee a tolerance for permanent occlusion. In this study, we evaluated the utility of computed tomography (CT) perfusion during BTO to quantify ischemic tolerance for detecting delayed ischemic stroke. MATERIALS AND METHODS: Forty-one patients who underwent BTO for the internal carotid artery were included. The correlations between the parameters of CT perfusion and collateral angiographic appearance or stump pressure during BTO were evaluated. The cerebral blood flow (CBF), cerebral blood volume, mean transit time (MTT), and time to peak (TTP) were obtained through CT perfusion, and the asymmetry ratios were determined. Collateral angiographic appearances were categorized into 5 grades (0-4). RESULTS: The collateral angiographic appearance showed moderate correlations with CBF, MTT, and TTP that was significant. Of these, the absolute value of the correlation coefficient was the highest for MTT. MTT also showed a moderate correlation with stump pressure. CBF and MTT were significantly different between the poor collateral group (grades 2 and 3) and the good collateral group (grade 4). Based on the MTT, the good collateral group was identified with high sensitivity (75.0%) and specificity (81.2%). CONCLUSIONS: In BTO, the MTT obtained through CT perfusion showed a correlation with collateral angiographic appearance and stump pressure. Thus, the MTT might be useful to quantify ischemic tolerance for detecting delayed ischemic stroke.
Subject(s)
Balloon Occlusion , Brain Ischemia/diagnostic imaging , Carotid Artery, Internal/diagnostic imaging , Cerebral Angiography , Cerebrovascular Circulation , Collateral Circulation , Computed Tomography Angiography , Intracranial Aneurysm/diagnostic imaging , Perfusion Imaging/methods , Adolescent , Adult , Aged , Blood Flow Velocity , Brain Ischemia/physiopathology , Carotid Artery, Internal/physiopathology , Child , Female , Humans , Intracranial Aneurysm/physiopathology , Male , Middle Aged , Predictive Value of Tests , Retrospective Studies , Young AdultABSTRACT
Monocarboxylate transporter 8 (MCT8) facilitates T3 uptake into cells. Mutations in MCT8 lead to Allan-Herndon-Dudley syndrome (AHDS), which is characterized by severe psychomotor retardation and abnormal thyroid hormone profile. Nine uncharacterized MCT8 mutations in Japanese patients with severe neurocognitive impairment and elevated serum T3 levels were studied regarding the transport of T3. Human MCT8 (hMCT8) function was studied in wild-type (WT) or mutant hMCT8-transfected human placental choriocarcinoma cells (JEG3) by visualizing the locations of the proteins in the cells, detecting specific proteins, and measuring T3 uptake. We identified 6 missense (p.Arg445Ser, p.Asp498Asn, p.Gly276Arg, p.Gly196Glu, p.Gly401Arg, and p.Gly312Arg), 2 frameshift (p.Arg355Profs*64 and p.Tyr550Serfs*17), and 1 deletion (p.Pro561del) mutation(s) in the hMCT8 gene. All patients exhibited clinical characteristics of AHDS with high free T3, low-normal free T4, and normal-elevated TSH levels. All tested mutants were expressed at the protein level, except p.Arg355Profs*64 and p.Tyr550Serfs*17, which were truncated, and were inactive in T3 uptake, excluding p.Arg445Ser and p.Pro561del mutants, compared with WT-hMCT8. Immunocytochemistry revealed plasma membrane localization of p.Arg445Ser and p.Pro561del mutants similar with WT-hMCT8. The other mutants failed to localize in significant amount(s) in the plasma membrane and instead localized in the cytoplasm. These data indicate that p.Arg445Ser and p.Pro561del mutants preserve residual function, whereas p.Asp498Asn, p.Gly276Arg, p.Gly196Glu, p.Gly401Arg, p.Gly312Arg, p.Arg355Profs*64, and p.Tyr550Serfs*17 mutants lack function. These findings suggest that the mutations in MCT8 cause loss of function by reducing protein expression, impairing trafficking of protein to plasma membrane, and disrupting substrate channel.
Subject(s)
Cell Membrane/metabolism , Cytoplasm/metabolism , Mental Retardation, X-Linked/genetics , Monocarboxylic Acid Transporters/genetics , Muscle Hypotonia/genetics , Muscular Atrophy/genetics , Protein Transport/genetics , Triiodothyronine/metabolism , Adolescent , Asian People , Cell Line, Tumor , Child , Child, Preschool , Genetic Vectors , Humans , Immunohistochemistry , In Vitro Techniques , Infant , Japan , Loss of Function Mutation , Male , Mental Retardation, X-Linked/metabolism , Mental Retardation, X-Linked/physiopathology , Monocarboxylic Acid Transporters/metabolism , Muscle Hypotonia/metabolism , Muscle Hypotonia/physiopathology , Muscular Atrophy/metabolism , Muscular Atrophy/physiopathology , Mutation , Symporters , Thyrotropin/metabolism , Thyroxine/metabolism , Transfection , Young AdultABSTRACT
Recent advances in computed tomography angiography (CTA) enable repeated imaging follow up for post-clipping surgery. The purpose of this study was to clarify the critical volume and configuration of the aneurysmal clip in the postoperative evaluation using volume rendering (VR) imaging, and present four-dimensional (4D)-CTA for these larger metal artifacts. A total of 44 patients with cerebral aneurysm, treated using clipping surgery, were included in this study. The metal artifact volume was assessed using CTA and the association between the type of clips and its metal artifact volume was analyzed. A VR image and a 4D-CTA were then produced, and the diagnostic accuracy of arteries around the clip or residual aneurysm on these images was evaluated. In the receiver operating characteristic (ROC) curve analysis, the cutoff value for metal artifacts was 2.32 mm3 as determined through a VR image. Patients were divided into two groups. Group 1 included patients with a simple and small clip, and group 2 included patients with multiple, large or fenestrated clips. The metal artifact volume was significantly larger in group 2, and the group incorporated the cutoff value. Post-clipping status on the VR image was significantly superior in group 1 compared with group 2. In group 2, the imaging quality of post-clipping status on 4D-CTA was superior in 92.9% of patients. The metal artifact volume was dependent on the number, size, or configuration of the clip used. In group 2, evaluation using a 4D-CTA eliminated the effect of the metal artifacts.
Subject(s)
Cerebral Angiography/methods , Four-Dimensional Computed Tomography/methods , Intracranial Aneurysm/diagnostic imaging , Intracranial Aneurysm/surgery , Neurosurgical Procedures/methods , Adolescent , Adult , Aged , Aged, 80 and over , Artifacts , Computed Tomography Angiography , Female , Humans , Male , Metals , Middle Aged , Postoperative Period , ROC Curve , Surgical Instruments , Virtual Reality , Young AdultABSTRACT
Cole-Carpenter syndrome is a rare skeletal dysplasia associated with low-bone mass or an osteogenesis imperfecta (OI)-like syndrome. Only 3 and 6 variants in SEC24D have been reported in patients with Cole-Carpenter syndrome type 2 and autosomal recessive OI, respectively. We describe a 15-year-old Japanese boy with short stature of the short-trunk type and craniofacial abnormalities including ocular proptosis, marked frontal bossing, midface hypoplasia, and micrognathia. These features were consistent with a diagnosis of Cole-Carpenter syndrome. He had low-bone mineral density and basilar impression. Whole exome sequencing analysis identified biallelic variants in SEC24D (p.Arg484* and p.Arg313His) in the patient. We will report a patient with compound heterozygous variants of SEC24D causing Cole-Carpenter syndrome type 2.
Subject(s)
Craniosynostoses/diagnosis , Craniosynostoses/genetics , Eye Abnormalities/diagnosis , Eye Abnormalities/genetics , Heterozygote , Hydrocephalus/diagnosis , Hydrocephalus/genetics , Mutation , Osteogenesis Imperfecta/diagnosis , Osteogenesis Imperfecta/genetics , Vesicular Transport Proteins/genetics , Adolescent , Alleles , Brain/abnormalities , Brain/diagnostic imaging , DNA Mutational Analysis , Genetic Association Studies , Humans , Japan , Male , Phenotype , Exome SequencingABSTRACT
Computed tomography angiography (CTA) is often used to assess the vascular status in moyamoya disease. The purpose of the study is to identify the characteristics of cortical arteries (M4) of moyamoya disease on CTA; the clinical significance of which is also discussed. A total of 38 hemispheric sides of 27 patients with moyamoya disease were included in this study. The number of M4 was visualized on CTA using cortical surface imaging and compared between the moyamoya disease group and the non-moyamoya disease group or the control group. Then, the clinical and radiological factors associated with the number of M4, the distribution of M4, and collateral circulation were examined. The number of M4 was lower in the moyamoya disease group than in the non-moyamoya disease group and in the control group (p < 0.05). There are few predictive clinical factors of the number of M4 except male sex. The prefrontal artery, precentral artery, central artery, and angular artery had a significantly higher prevalence in moyamoya disease (p < 0.05). The durocortical and periventricular anastomosis had a significantly higher prevalence in moyamoya disease (p < 0.05). The prevalence and distribution pattern of cortical arteries in moyamoya disease differed from that of the non-moyamoya disease group, and the distribution patterns of M4 might be influenced by collateral circulation. It is thus essential to recognize M4 to assess the recipient artery so as to ensure superficial temporal artery-middle cerebral artery bypass.
Subject(s)
Brain/blood supply , Brain/diagnostic imaging , Middle Cerebral Artery/diagnostic imaging , Middle Cerebral Artery/surgery , Moyamoya Disease/diagnostic imaging , Moyamoya Disease/surgery , Adolescent , Adult , Aged , Aged, 80 and over , Cerebrovascular Circulation , Child , Child, Preschool , Circle of Willis/diagnostic imaging , Circle of Willis/surgery , Computed Tomography Angiography , Female , Humans , Magnetic Resonance Angiography , Male , Middle Aged , Multidetector Computed Tomography , Tomography, Emission-Computed, Single-Photon , Vascular Surgical Procedures , Young AdultABSTRACT
Aseptic bone flap resorption, a rare complication after cranioplasty following decompressive craniectomy, is more likely to develop in children. We experienced two cases of aseptic bone flap resorption and identified potential pathophysiological mechanisms through histological findings. In the first case, an 11-year-old girl underwent decompressive craniectomy due to brain swelling with contusion. An autologous bone flap was cryopreserved for four months. Twenty-five months after cranioplasty with autologous bone flap, aseptic bone flap resorption was observed, and cranioplasty was performed with ceramic bone. Most of the histological findings in this case showed mature osseous tissue, while some showed osteoclasts and new bone formation due to endochondral ossification. In the second case, a 10-year-old girl underwent frontal craniectomy and removal of contusional hematoma. Fourteen months after cranioplasty with autologous bone flap, aseptic bone flap resorption was observed, and cranioplasty was performed with ceramic bone paste. The progression of bone flap resorption was not recognized for 12 months. In these cases, new bone formation was not necessarily linked to pathological bone flap resorption. It is supposed that the balance between bone destruction and new bone formation was disrupted by unknown factors.
Subject(s)
Brain Edema/surgery , Brain Injuries/surgery , Skull Fractures/surgery , Surgical Flaps , Brain Edema/etiology , Brain Injuries/diagnostic imaging , Child , Decompressive Craniectomy , Female , Hematoma/etiology , Hematoma/surgery , Humans , Reoperation , Skull Fractures/diagnostic imaging , Tomography, X-Ray ComputedABSTRACT
BACKGROUND: White matter lesions (WMLs) indicate progressive cerebral small vessel disease and are frequently observed in aging people and stroke patients. The purpose of this study was to evaluate WMLs and the influence of revascularization surgery for WMLs in patients with moyamoya disease. METHODS: A total of 42 hemispheres in 21 patients with moyamoya disease were included in this analysis. WMLs were detected on fluid-attenuated inversion recovery (FLAIR) imaging in 24 hemispheres. The distribution pattern of WMLs was categorized. WMLs were measured by manually creating a region of interest, and the total WML volume in the hemisphere was estimated and compared with the control group. Clinical features of patients with WMLs and postoperative changes were analyzed. RESULTS: The total WML volume in moyamoya disease was significantly higher than that of controls (P = .014). WMLs were observed in the internal watershed zone, especially in the watershed zone between the anterior cerebral artery and the middle cerebral artery. Patients with WMLs had significantly higher magnetic resonance angiography scores compared with controls (P = .015). A given patient's total WML volume per hemisphere was significantly decreased after surgery (P = .001). CONCLUSIONS: WMLs on FLAIR imaging increase in patients with moyamoya disease and are reversible by revascularization surgery. Improvement of cerebral blood flow is partly related to the decrease in WMLs. The present results provide insight into the potential role of revascularization surgery.
Subject(s)
Leukoencephalopathies/etiology , Magnetic Resonance Imaging , Moyamoya Disease/surgery , Neurosurgical Procedures , White Matter/diagnostic imaging , Adolescent , Adult , Child , Female , Humans , Leukoencephalopathies/diagnostic imaging , Magnetic Resonance Angiography , Male , Middle Aged , Moyamoya Disease/complications , Moyamoya Disease/diagnostic imaging , Perfusion Imaging/methods , Predictive Value of Tests , Remission Induction , Retrospective Studies , Time Factors , Tomography, Emission-Computed, Single-Photon , Treatment Outcome , Young AdultABSTRACT
OBJECT: We report a relatively rare case of pediatric iatrogenic extradural vertebral arteriovenous fistula. CASE: A five-year-old boy with a history of multiple surgeries for Total Anomalous Pulmonary Venous Return(TAPVR)exhibited vascular engorgement of his right neck from about 4 years old. We thought that this was caused by his past operations. He demonstrated swollen blood vessels in the right neck and pulmonary hypertension with increased right heart load. A right extradural vertebral arteriovenous fistula was seen on angiography. A high flow shunt was present at the V1 segment, at the level of the sixth cervical vertebra, and a 12 mm venous pouch was present anterior to the vertebral artery. We recognized the outlet passages of the cranial tract were the vertebral venous plexus, internal jugular vein, and right atrium. We performed endovascular transarterial embolization using a hydrogel coil. As a result, we treated with a high volume embolization ratio. Following treatment, the arteriovenous fistula disappeared while anterograde blood flow of the vertebral artery was preserved. The patient demonstrated no neurological deficits, improved right heart load, and his venous pouch of the neck. He has had no recurrence in the two years after treatment. CONCLUSION: Endovascular surgery is effective and safe for the treatment of iatrogenic vertebral arteriovenous fistula.
Subject(s)
Arteriovenous Fistula/therapy , Embolization, Therapeutic/methods , Angiography , Arteriovenous Fistula/diagnostic imaging , Catheterization, Peripheral , Child, Preschool , Humans , Iatrogenic Disease , Imaging, Three-Dimensional , Male , Tomography, X-Ray Computed , Vertebral Artery/diagnostic imagingABSTRACT
BACKGROUND: This study was undertaken to retrospectively investigate clinical features of subarachnoid hemorrhage (SAH) with cardiopulmonary arrest in patients achieving return of spontaneous circulation (ROSC) in order to explore the possibility of long-term survival. METHODS: Of 143 SAH patients with cardiopulmonary arrest in our hospital between April 2004 and June 2012, data on 59 (41%) patients who attained ROSC were analyzed to determine the predictive factors for neurologic recovery and outcome. Recovery of brainstem reflexes and improvement of Glasgow Coma Scale (GCS) motor score were noted (postresuscitation neurologic restorative assessment, grade I) in 5, and 2 of these patients survived. RESULTS: By-grade analysis of patient background characteristics revealed a significantly shorter duration of cardiac arrest (P = .001) and a significantly smaller adrenaline dose (P = .011) for grade I patients. A logistic analysis of 1-week survival data revealed significant differences in duration of cardiac arrest (P = .022) and adrenaline dose (P = .019), with odds ratios of 0.89 and 0.25, respectively. Cox regression analysis of mortality data revealed significant differences in the duration of cardiac arrest (P = .012), adrenaline dose (P < .0001), and location of ROSC (P = .016), with hazard ratios of 1.03, 1.43, and 1.98, respectively. CONCLUSIONS: Cardiac arrest caused by SAH is a disease state with a grave prognosis, but there is the possibility of a good survival outcome when the administration of a small dose of adrenaline results in the rapid recovery of brainstem reflexes.
Subject(s)
Cardiopulmonary Resuscitation , Out-of-Hospital Cardiac Arrest/therapy , Subarachnoid Hemorrhage/therapy , Survivors , Adrenergic Agonists/administration & dosage , Adult , Aged , Brain Stem/drug effects , Brain Stem/physiopathology , Cardiopulmonary Resuscitation/adverse effects , Cardiopulmonary Resuscitation/mortality , Cerebral Angiography/methods , Disability Evaluation , Echocardiography , Epinephrine/administration & dosage , Female , Glasgow Coma Scale , Humans , Logistic Models , Male , Middle Aged , Odds Ratio , Out-of-Hospital Cardiac Arrest/diagnosis , Out-of-Hospital Cardiac Arrest/etiology , Out-of-Hospital Cardiac Arrest/mortality , Out-of-Hospital Cardiac Arrest/physiopathology , Proportional Hazards Models , Reflex/drug effects , Retrospective Studies , Risk Assessment , Risk Factors , Subarachnoid Hemorrhage/complications , Subarachnoid Hemorrhage/diagnosis , Subarachnoid Hemorrhage/mortality , Subarachnoid Hemorrhage/physiopathology , Time Factors , Tomography, X-Ray Computed , Treatment OutcomeABSTRACT
Arterial spin labeling (ASL) magnetic resonance imaging (MRI) is a technique for depicting cerebral perfusion without contrast medium. The purpose of this study was to determine whether ASL can be used to detect hyperperfusion after revascularization for moyamoya disease as effectively as N-isopropyl-[123I]ß-iodoamphetamine ((123)I-IMP) single-photon emission computed tomography (SPECT). Fifteen consecutive patients with moyamoya disease were included in the study. All patients underwent surgical revascularization. Postoperatively, regional cerebral blood flow (rCBF) was measured by flow-sensitive alternating inversion recovery (FAIR) ASL and (123)I-IMP SPECT during the acute stage, and rCBF of the operative side was compared with the other side. The asymmetry ratio (AR) was then calculated from the rCBF as measured using each modality. The postoperative AR of ASL was moderately correlated with that of (123)I-IMP SPECT (y = 0.180x + 0.819; R = 0.80; P = .0003). In this series, 2 patients (13.3%) suffered symptomatic hyperperfusion after revascularization and accordingly exhibited increased AR of ASL. Our data indicate that early increases in rCBF in patients with hyperperfusion could be detected using FAIR ASL supplemental to (123)I-IMP SPECT after revascularization. Our data indicate that FAIR ASL is a convenient method for evaluating hyperperfusion that can be performed repeatedly without the use of contrast medium or radioisotopes.
Subject(s)
Cerebral Arteries/surgery , Cerebral Revascularization , Cerebrovascular Circulation , Magnetic Resonance Imaging , Moyamoya Disease/surgery , Myocardial Perfusion Imaging/methods , Spin Labels , Adolescent , Adult , Blood Pressure , Brain Edema/diagnosis , Brain Edema/physiopathology , Cerebral Arteries/diagnostic imaging , Cerebral Arteries/physiopathology , Cerebral Revascularization/adverse effects , Child , Child, Preschool , Female , Humans , Iofetamine , Magnetic Resonance Angiography , Male , Middle Aged , Moyamoya Disease/diagnosis , Moyamoya Disease/physiopathology , Predictive Value of Tests , Radiography , Radiopharmaceuticals , Time Factors , Tomography, Emission-Computed, Single-Photon , Treatment Outcome , Young AdultABSTRACT
The treatment of a large-giant aneurysm in the cavernous portion of an internal carotid artery(ICA)requires occlusion of the ICA, with or without extracranial-intracranial(EC-IC)bypass surgery. Although a balloon occlusion test(BOT)is used to determine the need for bypass surgery and select the bypass type, the criteria are not well established. In this study, we retrospectively analyzed 10 consecutive patients(11 sides)with cavernous large and giant aneurysms treated during the past 8 years. Therapeutic strategies for each patient were selected according to the results of neurological and radiological examinations, and regional cerebral oxygen saturation. A total of 6 high-flow bypasses were placed before ICA occlusions. Three patients had STA-MCA bypasses before ICA occlusions, and two underwent endovascular ICA occlusion without bypass surgery. Favorable outcomes were obtained in all cases with respect to cranial nerve palsy, but one patient had insufficient ipsilateral cerebral blood flow postoperatively, and hemiparesis was revealed. An emergent STA-MCA bypass was performed in this case, which minimized the hemiparesis. Despite radiological evaluation during the BOT procedures, we had one false negative result. This indicates that the BOT requires increased sensitivity and specificity, and that a safety margin should be adopted when determining indications for bypass surgeries.
Subject(s)
Carotid Artery Diseases/surgery , Carotid Artery, Internal/surgery , Intracranial Aneurysm/surgery , Adolescent , Adult , Aged , Balloon Occlusion/methods , Brain/blood supply , Carotid Artery Diseases/diagnosis , Cerebral Revascularization/methods , Cerebrovascular Circulation , Child , Humans , Image Processing, Computer-Assisted , Intracranial Aneurysm/diagnosis , Male , Middle Aged , Retrospective Studies , Treatment Outcome , Young AdultABSTRACT
BACKGROUND: An intratumoral aneurysm encased within the associated intracranial tumor is rare, and hemorrhage caused by its rupture is even more rare. While urgent and adequate surgical treatment is important, the treatment can be difficult given the limited understanding of this rare condition. OBSERVATIONS: A 69-year-old man who had undergone meningioma surgery 30 years prior presented with a disturbance in consciousness. Magnetic resonance imaging revealed massive intracerebral and subarachnoid hemorrhage. A round, partially calcified mass, which was diagnosed as recurrent meningioma, was also observed. Subsequent cerebral angiography revealed that the source of the hemorrhage was an intratumoral aneurysm in the dorsal internal carotid artery (ICA) encased within the recurrent meningioma. Urgent surgical ICA trapping and high-flow graft bypass were conducted. The postoperative course was uneventful, and he was referred to another hospital for rehabilitation. LESSONS: This is the first case report of a ruptured intratumoral aneurysm being treated with urgent combined revascularization and parent artery trapping surgery. This surgical approach may be a feasible treatment option for such a challenging condition. Additionally, this case highlights the importance of diligent long-term follow-up after skull-base surgery, as minor intraoperative vascular wall injury may trigger the development and rupture of an intracerebral aneurysm.
ABSTRACT
Objective: Recent studies evaluating plaque protrusion at carotid artery stenting (CAS) using optical coherence tomography showed not a few cases of plaque protrusion when using double-layer micromesh stents. We report a case of symptomatic internal carotid artery (ICA) stenosis with at-risk unstable plaques in which CAS was successfully performed using a stent-in-stent technique by the combined use of a closed-cell stent and a dual-layer micromesh stent. Case Presentation: An 87-year-old Japanese man with dysarthria and right hemiparesis was diagnosed with atheromatous cerebral embolism caused by severe left ICA stenosis on MRI and DSA. MRI with T1-weighted black blood methods showed high intensities in the plaques of the left ICA, suggesting unstable plaque characteristics with intraplaque hemorrhage components. On day 20, CAS was performed. After the pre-stent dilation under proximal and distal protection, a Carotid WALLSTENT was placed to cover the stenotic lesion. Then, a CASPER Rx was placed from the proximal left ICA to the common carotid artery to cover the Carotid WALLSTENT. Although visible plaque debris was recognized in the aspirated blood, the debris became invisible after aspiration of 1300 mL. Postoperative angiography showed enough dilation of the left ICA, with no plaque protrusion or acute stent thrombosis. The patient had an uneventful postoperative course and was discharged without any neurological sequelae. Conclusion: The present case suggests that the combined stent-in-stent technique using a closed-cell stent and a micromesh stent can be considered as one of the treatment strategies for preventing plaque protrusion and procedural ischemic complications in patients with high-risk carotid plaques.
ABSTRACT
OBJECTIVE: Clinical characteristics of endovascular treatment (EVT) for acute ischemic stroke (AIS) secondary to atherosclerosis are not fully delineated. An optimal treatment strategy with considerations of stroke etiology has not yet been established. Here-in, we performed retrospective analysis of EVT for atherosclerotic AIS. METHODS: Data from patients with AIS who underwent EVT between 2017 and 2022 were analyzed. Clinical characteristics, procedural data, and outcomes were assessed. Further analysis was conducted to elucidate the factors associated with clinical outcomes. And data of patients with poor clinical outcomes (mRS, 5 or 6) were evaluated further to determine the primary cause. RESULTS: Among 194 patients who received EVT, 40 (20.6%) were diagnosed with AIS with an atherosclerotic etiology. The rates of successful reperfusion (TICI 2b or 3) and good clinical outcomes (mRS, 0-2) were 95.0% and 45.0%, respectively. No procedure-related complications were noted. Older age (p = 0.007), more severe baseline NIHSS score (p = 0.004), lesion in the posterior circulation (p = 0.025), and recanalization failure (p = 0.027) were more frequently observed in patients with poor clinical outcomes. Brainstem infarction and postprocedural intracerebral hemorrhage were the main reasons for poor clinical outcomes. CONCLUSION: The EVT for atherosclerotic AIS were effective and safe. Older age, more severe NIHSS score, lesions in the posterior circulation, and recanalization failure were the factors associated with poor clinical outcomes. It is important to recognize that these factors may aggravate the clinical response to this promising therapy, even in patient successful recanalization was attained.
Subject(s)
Atherosclerosis , Brain Ischemia , Endovascular Procedures , Ischemic Stroke , Stroke , Humans , Ischemic Stroke/diagnostic imaging , Ischemic Stroke/etiology , Ischemic Stroke/surgery , Retrospective Studies , Treatment Outcome , Endovascular Procedures/adverse effects , Stroke/etiology , Stroke/surgery , Atherosclerosis/complications , Thrombectomy/adverse effects , Brain Ischemia/diagnostic imaging , Brain Ischemia/etiology , Brain Ischemia/surgeryABSTRACT
Microsurgical resection remains an important treatment for cerebral arteriovenous malformations (AVMs). We developed an accurate method for planning AVM resections using multidetector row CT (MDCT). Between January 2007 and January 2011, 21 consecutive patients with AVMs were enrolled in this study. Sixteen patients were symptomatic, and of these, 15 had a hemorrhagic onset. Preoperative CT angiography (CTA) was performed using an MDCT scanner (GE Lightspeed VCT; GE Healthcare, Milwaukee, WI, USA). In total, 1 to 1.2 mL/kg of iopamidol, a low-osmolar iodinated contrast material, was administered intravenously with the bolus tracking method. In all cases, three-dimensional CTA imaging demonstrated critical arterial feeders and their specific geometric associations with the nidus. Accurate visualization of the architecture of AVMs and surgical trajectory was possible with the volume rendering method, especially when using transparency imaging. Additionally, employing feeder extraction imaging with segmentation post-processing, clear preoperative identification of the feeding arteries around the surrounding structures allowed for not only intraoperative orientation but also planning for presurgical embolization. Moreover, the precise anatomical structures of the brain surface were obtained using fusion imaging with MRI. While it is difficult to scientifically assign a value to a technology, we sought to objectively assess the utility of the currently available CTA. The significant benefits of this modality for presurgical planning include enhanced surgeon confidence and optimization of the sequence of surgical procedures.
Subject(s)
Intracranial Arteriovenous Malformations/diagnostic imaging , Tomography, X-Ray Computed , Adolescent , Adult , Angiography, Digital Subtraction/methods , Cerebral Angiography/methods , Child , Child, Preschool , Embolization, Therapeutic/methods , Female , Humans , Imaging, Three-Dimensional/instrumentation , Infant , Intracranial Arteriovenous Malformations/surgery , Magnetic Resonance Imaging , Male , Middle Aged , Neuronavigation/instrumentation , Neuronavigation/methods , Young AdultABSTRACT
Perimesencephalic subarachnoid hemorrhage (PMSAH) is a group of diseases characterized by bleeding around the brainstem. In recent years, it has been suggested that PMSAH is associated with the venous system. We report a case of PMSAH caused by stenosis of the junction of the vein of Galen (VG) and the rectus sinus (RS). A 39-year-old man presented with restlessness at work. He was administered diazepam, and a few minutes later, his consciousness became clear. Imaging showed subarachnoid hemorrhage (SAH) around the right midbrain, occlusion of the right sigmoid sinus and the right transverse sinus, stenosis of the junction of the VG and RS, T2 hyperintensity in the right middle temporal gyrus, and obstruction of the right vein of Labbe. The location of the SAH coincided with stenosis of the junction of the VG and RS. PMSAH was diagnosed due to the increase in intracranial venous pressure caused by the patient's sinus obstruction in addition to the stenosis of the junction of the VG and RS. It is necessary to pay attention to venous return when PMSAH is observed.
ABSTRACT
Therapeutic hypothermia for severe traumatic brain injury (TBI) has been repeatedly studied, but no past studies have assessed the detailed head computed tomography (CT) findings. We sought to investigate individual CT findings of severe TBI patients treated with targeted temperature management utilizing the head CT database obtained from the Brain Hypothermia study. Enrolled patients underwent either mild therapeutic hypothermia (32.0°C-34.0°C) or fever control (35.5°C-37.0°C). We assessed individual head CT images on arrival and after rewarming and investigated the correlations with outcomes. The initial CT data were available for 125 patients (hypothermia group = 80, fever control group = 45). Baseline characteristics and CT findings, such as hematoma thickness and midline shift, were similar in all aspects between the two groups. The favorable outcomes in the hypothermia and fever control groups were 38 (47.5%) and 24 (53.3%; p = 0.53) for all 125 patients, respectively; 21 (46.7%) vs. 10 (38.5%; p = 0.50) for 71 patients with acute subdural hematoma (SDH), respectively; and 12 (75.0%) vs. 4 (36.4%; p = 0.045) in 27 young adults (≤50 years) with acute SDH, respectively. There was a trend toward favorable outcomes for earlier time to reach 35.5°C (190 vs. 377 min, p = 0.052) and surgery (155 vs. 180 min, p = 0.096) in young patients with acute SDH. The second CT image revealed progression of the brain injury. This study demonstrated the potential benefits of early hypothermia in young patients with acute SDH, despite no difference in CT findings between the two groups. However, the small number of cases involved hindered the drawing of definitive conclusions. Future studies are warranted to validate the results.