[Preoperative Diagnosis of Spinal Immature Teratoma Associated with Cerebrospinal Fluid Leakage from the Congenital Dermal Sinus Tract in a 0-Day-Old Infant:A Case Report].

We report the case of a patient with spinal immature teratoma and cerebrospinal fluid leakage from the congenital dermal sinus tract. A 0-day-old female infant presented with a subcutaneous soft mass with a dimple in the lumbosacral region at birth. Magnetic resonance imaging revealed a mixed low-intensity mass located in the extraspinal and intraspinal canal with a sinus tract. The reconstructed three-dimensional spinal computed tomography image showed spina bifida and ectopic ossification at the dorsal aspect of the sacrum. Urgent removal of the tumor and dermal sinus tract was then performed under evoked electromyography monitoring. The resected tumor was histopathologically diagnosed as immature teratoma.

Camurati-Engelmann disease combined with transethmoidal meningoencephalocele: illustrative case.

BACKGROUND: Camurati-Engelmann disease (CED) is a rare disorder characterized by progressive cranial hyperostosis and diaphyseal sclerosis of the long bones. Chronic intracranial hypertension gradually occurs due to progressive cranial vault hyperostosis. OBSERVATIONS: A 57-year-old man who had been diagnosed with CED at 9 years old suddenly developed cerebrospinal fluid rhinorrhea. A bone defect of the right cribriform plate and protrusion of brain tissue from the right cribriform plate into the right nasal cavity were identified. The patient underwent endoscopic resection of the meningoencephalocele combined with the bath-plug procedure. After surgery, cerebrospinal fluid rhinorrhea disappeared. LESSONS: Chronic intracranial hypertension due to progressive cranial vault hyperostosis in CED may cause a bone defect and meningoencephalocele in the anterior skull base, resulting in cerebrospinal fluid rhinorrhea.

Predicting the consistency of intracranial meningiomas using apparent diffusion coefficient maps derived from preoperative diffusion-weighted imaging.

OBJECTIVE: The consistency of meningiomas is a critical factor affecting the difficulty of resection, operative complications, and operative time. The apparent diffusion coefficient (ADC) is derived from diffusion-weighted imaging (DWI) and is calculated using two optimized b values. While the results of comparisons between the standard ADC and the consistency of meningiomas vary, the shifted ADC has been reported to be strongly correlated with liver stiffness. The purpose of the present prospective cohort study was to determine whether preoperative standard and shifted ADC maps predict the consistency of intracranial meningiomas. METHODS: Standard (b values 0 and 1000 sec/mm2) and shifted (b values 200 and 1500 sec/mm2) ADC maps were calculated using preoperative DWI in patients undergoing resection of intracranial meningiomas. Regions of interest (ROIs) were placed within the tumor on standard and shifted ADC maps and registered on the navigation system. Tumor tissue located at the registered ROI was resected through craniotomy, and its stiffness was measured using a durometer. The cutoff point lying closest to the upper left corner of a receiver operating characteristic (ROC) curve was determined for the detection of tumor stiffness such that an ultrasonic aspirator or scissors was always required for resection. Each tumor tissue sample with stiffness greater than or equal to or less than this cutoff point was defined as hard or soft tumor, respectively. RESULTS: For 76 ROIs obtained from 25 patients studied, significant negative correlations were observed between stiffness and the standard ADC (ρ = -0.465, p < 0.01) and the shifted ADC (ρ = -0.490, p < 0.01). The area under the ROC curve for detecting hard tumor (stiffness ≥ 20.8 kPa) did not differ between the standard ADC (0.820) and the shifted ADC (0.847) (p = 0.39). The positive predictive value (PPV) for the combination of a low standard ADC and a low shifted ADC for detecting hard tumor was 89%. The PPV for the combination of a high standard ADC and a high shifted ADC for detecting soft tumor (stiffness < 20.8 kPa) was 81%. CONCLUSIONS: A combination of standard and shifted ADC maps derived from preoperative DWI can be used to predict the consistency of intracranial meningiomas.

Hypereosinophilia with systemic thrombophlebitis.

A 34-year-old Japanese woman developed subcutaneous induration in the left thigh, then showed extreme eosinophilia, and died of hemorrhagic infarction of the brain. Autopsy revealed endocarditis with eosinophil infiltration and systemic thrombophlebitis, including pulmonary veins and intrahepatic branches of the portal vein. Arterial structure was relatively preserved. She had no clinical history of asthma and had anti-ascarid IgE antibody at postmortem serological examination; thus, her disease does not fulfill the diagnostic criteria of Churg-Strauss syndrome and idiopathic hypereosinophilic syndrome (HES). Her organ involvement is, however, consistent with that of HES; thus, her pathophysiological conditions would resemble those of HES. Systemic thrombophlebitis without arterial lesion in patients with hypereosinophilia has never been reported, and this case would broaden the spectrum of vascular lesions in these patients.

Recovery process of immediate prolonged posttraumatic coma following severe head injury without mass lesions.

The recovery process of immediate posttraumatic coma was investigated in 24 patients with severe head injury. The correlation between poor outcome in the recovery process and magnetic resonance (MR) imaging findings was analyzed. MR imaging was performed within the first 7 days for all patients. The recovery process was classified into phase 1 for recovery to moderately disabled and phase 2 to good recovery (GR) according to the Glasgow Outcome Scale. The median of phase 1 was 21.0 days. Four patients did not recover to GR and had poor outcome. Twenty patients recovered to GR. Thirteen patients had short phase 2 of under 10 days and seven patients had long phase 2 of over 60 days. All patients had abnormal lesions on MR imaging considered to be diffuse axonal injury. The number of lesions ranged from two to 10, with a mean of five. Lesions in the dorsal upper brainstem were significantly associated with poor outcome (p < 0.05). The combination of focal lesions in the callosal splenium and dorsal upper brainstem was most common in patients with poor outcome. Patients with long phase 2 had significantly more lesions than patients with short phase 2.

[A case of the development of cavum vergae after head trauma].

We report a case of the development of cavum septi pellucidi and cavum Vergae after head trauma in a 29-year-old female patient. After the accident, cavum septi pellucidi and cavum Vergae gradually expanded. Using magnetic resonance imaging, we followed up the patient for 33 months after the head trauma. Preoperatively, metrizamide CT cisternography was performed in order to investigate CSF flow between the cavum septi pellucidi/cavum Vergae and the subarachnoid space. Using neuroendoscopy, we succeeded in opening the wall of the cavum septi pellucidi via anterior horn of the right lateral ventricle. Cystography was performed during the operation. The volume of the cavum septi pellucidi/cavum Vergae decreased remarkably. In this case, our CSF flow studies revealed that CSF flowed into the posterior part of the cavum Vergae from the third ventricle and did not flow backward to the third ventricle. Therefore, we considered that the development of cavum septi pellucidi/cavum Vergae was related to a one-way valve mechanism between the posterior part of the cavum Vergae and the third ventricle.

Growth hormone-producing pituitary adenoma regrowing as pituitary adenoma with neuronal choristoma 14 years after tumor removal.

BACKGROUND: We report a case of an uncommon association of pituitary adenoma with neuronal choristoma that usually is diagnosed at initial surgery. CASE DESCRIPTION: A 50-year-old woman with acromegaly and bitemporal hemianopsia underwent removal of a pituitary adenoma via the transsphenoidal approach. Histologic examination of the first surgical specimen demonstrated only adenoma, which was eosinophilic and expressed growth hormone. Fourteen years later, bitemporal hemianopsia recurred, and magnetic resonance imaging revealed regrowth of the residual tumor. DISCUSSION: The patient underwent removal of the regrown tumor via the transsphenoidal approach. Histologic examination of the second surgical specimen revealed gangliocytoma and a small component of pituitary adenoma. CONCLUSIONS: The present case report supports the theory that pituitary adenoma with neuronal choristoma might represent the result of neuronal differentiation from pituitary adenoma.

Combined measurement of cerebral and cerebellar blood flow on preoperative brain perfusion SPECT imaging predicts development of new cerebral ischemic events after endarterectomy for symptomatic unilateral cervical carotid stenosis.

PURPOSE: The aim of this study was to determine whether the ratio of blood flow contralateral-to-affected asymmetry in the cerebellar hemisphere to blood flow affected-to-contralateral asymmetry in the middle cerebral artery (MCA) territory (AR(cbl)/AR(MCA)) on preoperative brain perfusion SPECT could identify patients at risk for new cerebral ischemic events after carotid endarterectomy (CEA) for symptomatic unilateral cervical carotid stenosis. For the purposes of this study, new cerebral ischemic events included neurological deficits and cerebral ischemic lesions on diffusion-weighted MRI. METHODS: Brain blood flow was assessed using 123I-IMP SPECT in 101 patients. A region of interest was automatically placed in the bilateral MCA territories and in the bilateral cerebellar hemispheres using a 3-dimensional stereotaxic region-of-interest template, and the AR(cbl)/AR(MCA) was calculated. Diffusion-weighted MRI was performed within 3 days before and 24 hours after surgery. Patients were neurologically tested before induction of general anesthesia and after recovery from general anesthesia. RESULTS: New cerebral ischemic events after CEA were observed in 12 patients (12%). Multivariate analysis revealed that only high AR(cbl)/AR(MCA) was significantly associated with the development of new postoperative cerebral ischemic events (95% confidence interval, 1.945-8.452; P = 0.0070). The AR(cbl)/AR(MCA) provided 75% sensitivity, 84% specificity, and 39% positive and 96% negative predictive values in predicting development of new postoperative cerebral ischemic events. CONCLUSIONS: The AR(cbl)/AR(MCA) on preoperative brain perfusion SPECT could identify patients at risk for new cerebral ischemic events after CEA for unilateral cervical carotid stenosis.

High-resolution diffusion tensor imaging for the detection of diffusion abnormalities in the trigeminal nerves of patients with trigeminal neuralgia caused by neurovascular compression.

PURPOSE: To detect diffusion abnormalities in the trigeminal nerves of patients with trigeminal neuralgia (TN) caused by neurovascular compression (NVC) by using a high-resolution diffusion tensor imaging (HR-DTI) technique. METHODS: Thirteen patients with TN and 14 healthy controls underwent HR-DTI scanning. After extracting the trigeminal nerve using a tractography technique, we measured the fractional anisotropy (FA) and apparent diffusion coefficient (ADC), and compared the contralateral ratios (CR) of these parameters between the patients and controls, and correlated these ratios with the cross-sectional areas of the nerves. RESULTS: The CRs of the FA values for the trigeminal nerves of the patients (1.00±0.15) had significantly higher variance than those of healthy controls (1.00±0.05) (P<.05) and showed a positive correlation with the cross-sectional area of the nerves (r=0.81). In contrast, the CRs of the ADC values were not significantly different between the two groups (1.02±0.10 and 1.01±0.08, respectively) and had no significant correlation with cross-sectional area. CONCLUSION: HR-DTI can detect an alteration in the relative FA values of affected trigeminal nerves and a correlation with atrophic changes in patients with NVC-induced TN.

Ganglioneurocytoma: distinctive variant of central neurocytoma.

Reported herein is a case of central neurocytoma with differentiation toward ganglion cells, considered to be a typical case of ganglioneurocytoma. Tumor cells of various degree of differentiation toward ganglion cells were intermingled with typical neurocytoma cells in a fibrillary background, with transition of tumor cells from typical neurocytoma cells to differentiated ganglion cells evident throughout the tumor. The tumor cell nuclei were positive for NeuN. Fine granular positivity for synaptophysin was seen in the cytoplasm of the tumor cells, and background fibrils and the cytoplasm of some ganglioid cells were positive for neurofilament. Several cases of central neurocytoma with ganglioid cells have been reported, with some diagnosed as ganglioneurocytoma. However, histopathological details and persuasive figures have been lacking. It is considered that the diagnosis of ganglioneurocytoma should be applied to tumors displaying the following characteristics: (i) clinical aspects such as location, demarcation and growth rate consistent with neurocytoma; (ii) transition between neurocytoma cells and ganglion cells; and (iii) ganglioid cells distributed throughout the tumors.

Local elevation of the anti-inflammatory interleukin-10 in the pathogenesis of chronic subdural hematoma.

We investigated the relationship between inflammatory and anti-inflammatory cytokines in the pathogenesis of chronic subdural hematoma (CSDH) by measuring the plasma and subdural fluid levels of interleukin-6 (IL-6), interleukin-8 (IL-8), and interleukin-10 (IL-10). The levels of IL-6, IL-8 and IL-10 were measured in the subdural fluid obtained from 34 patients with CSDH, using the enzyme-linked immunosorbent assay. The patients were classified into a high IL-10 group and a low IL-10 group according to the level of IL-10 in their subdural fluid samples. The subdural fluid levels of IL-6 and IL-8 were significantly higher in the high IL-10 group than in the low IL-10 group (P<0.05). A tendency for the patients in the low IL-10 group to show the separated or layer type of pattern on the CT scans was noted.