Repeated shunt dysfunction due to Barium allergy that was difficult to differentiate from shunt infection.

We encountered a case of repeated shunt dysfunction caused by barium allergy. The patient was a 60-year-old male who underwent ventricular peritoneal shunting for hydrocephalus following subarachnoid hemorrhage due to a ruptured aneurysm;however, it malfunctioned many times. A patch test performed after the third reconstruction was positive for barium. To the best of our knowledge, this is the first case report of shunt malfunction due to barium allergy. The patch test is useful in cases of suspected allergy-related dysfunction. We recommend the introduction of barium into antigen testing using the patch test. J. Med. Invest. 70 : 521-523, August, 2023.

Clinical features of herpes simplex virus reactivation after microvascular decompression for trigeminal neuralgia: Experience of 200 patients and a literature review.

Background: Herpes simplex virus (HSV) reactivation occasionally develops in the early postoperative period after microvascular decompression (MVD) for trigeminal neuralgia (TN). Therefore, the present study investigated the clinical features of this phenomenon. Methods: The study cohort comprised 200 patients with 125 women aged between 17 and 90 years (median age, 66 years) who underwent MVD for TN between January 2010 and December 2020. Characteristics were compared between patients with and without HSV reactivation and clinical features were analyzed. Results: Twenty patients had HSV reactivation: herpes labialis in 18 and herpes zoster (final diagnosis) in 2. A multivariate analysis revealed independent correlations between postoperative HV reactivation and a previous history of herpes labialis (odds ratios [OR]: 6.32, P = 0.0003) and reoperation for recurrent or persistent pain (OR: 5.06, P = 0.0211). No significant differences were observed in pain relief, postoperative facial numbness, or Barrow Neurological Institute Pain Intensity/Facial Numbness Scores in the past follow-up between patients with and without HSV reactivation. HSV reactivation manifested at a median of the 4th postoperative day (1-10 days) and its location was not related to the preoperative distribution of facial pain. All patients were treated with local acyclovir and were completely cured within 1-2 weeks. Conclusion: HSV reactivation occurred in 10% of patients after MVD including 1% of herpes zoster. A previous history of herpes labialis and reoperation was identified as risk factors for reactivation. Symptoms were completely cured by antiviral drugs within 1-2 weeks. It is important to note that cases of herpes zoster may be confused with cases of HSV after MVD.

Venous Flow Conversion Technique for Sacrificing the Superior Petrosal Vein During Microvascular Decompression for Trigeminal Neuralgia.

BACKGROUND: Microvascular decompression for trigeminal neuralgia (TN) may require sacrifice of the superior petrosal vein (SPV), with potential risks of ischemia and hemorrhagic complications due to impaired venous return. OBJECTIVE: To investigate methods for safely sacrificing the SPV. METHODS: We retrospectively reviewed 21 cases in 346 consecutive microvascular decompression surgeries for TN. They were intraoperatively identified as SPV and its tributaries being the offending vessels causing TN and were intentionally sacrificed. RESULTS: The transverse pontine vein (TPV) was sacrificed in 10 patients. The main trunk of the SPV was sacrificed using the TPV as a collateral flow pathway in 10 patients. No complications occurred related to impaired venous return. CONCLUSION: The venous flow conversion technique can be applied to safely sacrificing the SPV and its tributaries with the TPV acting as a collateral blood flow pathway to prevent postoperative impaired venous return.

Resolution of the large middle fossa arachnoid cyst without any identifiable cause: Case report.

Although the possibility of spontaneous regression of intracranial arachnoid cysts (AC) during observational follow-up is widely recognized, the number of reports documenting such clinical course, often associated with the mild head trauma, is rather limited. We present a case of nearly complete resolution of the large middle fossa AC in a 5-year-old boy without any identifiable cause in 2.3 years after the initial diagnosis. It once again justifies observational strategy for AC not accompanying by mass effect and manifesting with minimal symptoms or diagnosed incidentally.

Acute Myocardial Infarction in a 17-year-old High-school Girl.

We report an unusual case of acute myocardial infarction in a high school girl. The patient was 17 years of age and had multiple coronary risk factors, including marked obesity with a body mass index (BMI) of 42.7 kg/m2, dyslipidemia and glucose intolerance. She had been an on and off smoker since she was 13 years of age. Due to the recent Westernization of the lifestyle, the prevalence of metabolic syndrome in the young generation has been increasing in Japan. Cardiovascular disease based on lifestyle-related diseases may become more common in young people.

[A Case of Segmental Arterial Mediolysis:Subarachnoid Hemorrhage Followed by Abdominal Bleeding].

We describe a case involving subarachnoid and intraperitoneal hemorrhage due to segmental arterial mediolysis(SAM). A 77-year-old female patient with sudden subarachnoid hemorrhage was immediately transferred to our institution. The hemorrhage was classified as grade 2 according to the World Federation of Neurosurgical Societies system. The patient was a non-smoker and did not drink alcohol regularly. A right internal carotid aneurysm was detected using CT angiography and was clipped during frontotemporal craniotomy. Bleeding was observed from the anterior wall of the internal carotid artery, and the tear was clipped. The patient had an uneventful postoperative course until sudden cardiopulmonary arrest eight days after craniotomy. She died of massive intraperitoneal hemorrhage. Autopsy revealed that the hemorrhage was due to dissection of the celiac artery. Tunica media denaturation was observed not only in the celiac artery, but also in the splenic and internal carotid arteries, which exhibited ruptured aneurysms, and the patient was diagnosed with segmental arterial mediolysis(SAM). SAM is an arterial degenerative disease affecting the medial layer of the arterial and dissecting walls. Multiple lesions are sometimes found. Radiographic imaging findings of SAM are similar to those of dissecting aneurysms, which are characterized by a single continuous dissection of the medial layer. As observed in this case, abdominal bleeding caused by SAM can occur after intracranial bleeding. When surgeons encounter unusual intracranial dissecting aneurysms, SAM should be considered as a differential diagnosis.

[Subarachnoid Hemorrhage of Unknown Origin at Initial Imaging Investigation].

In some patients with spontaneous subarachnoid hemorrhage(SAH), initial imaging investigations may not be able to detect a bleeding source;repeat imaging may be necessary to reveal these lesions. We reviewed a consecutive series of 45 patients with SAH and negative initial digital subtraction angiograms(DSA)during a 15-year period. The aims were to document the frequency and reason for the negative initial investigations, to determine the appropriate modality and timing of repeat examinations, and to investigate the identified bleeding sources. Twenty-eight(62%)patients underwent repeat DSA, 35(78%)underwent magnetic resonance imaging(MRI), and 33(73%)underwent computed tomography angiography(CTA). Nine lesions(5 small aneurysms, 2 craniocervical junction arteriovenous fistulas, 1 arteriovenous malformation, and 1 internal carotid artery dissection)were identified on subsequent DSA after 2-3 weeks. Most aneurysms were identified on an atypical vascular tree. CTA or MRI alone were unable to disclose the culprit lesions. In retrospect, human errors including oversight were the major reasons for the negative initial investigation results. It is, however, difficult to search for a tiny vascular lesion that might be anywhere in the cranium. Repeat DSA is still the gold standard for the inspection of hidden bleeding sources in patients with SAH of unknown origin.

[Endarterectomy for Internal Carotid Artery Occlusion, with an Aberrant Branch of the Internal Carotid Artery Maintaining Blood Flow Distal to the Complete Occlusion:A Case Report].

Doppler sonography accurately identifies occlusion of the internal carotid artery(ICA)and current surgical guidelines do not list an occluded ICA as an indication for carotid endarterectomy(CEA). We encountered an unusual case, for which we performed CEA. The left ICA was occluded by atherosclerosis, and was reconstituted via an aberrant branch of the occipital artery. A 71-year-old man was referred following brain infarction. Carotid duplex sonography(CDS)demonstrated occlusion of his left ICA, with flow in the distal ICA beyond the occlusion("Sandwich stump sign"). 3D computed tomography angiography and cervical angiography diagnosed ICA occlusion with flow in the distal ICA, and the patient underwent CEA. Careful evaluation is required when apparent occlusion of the ICA is detected to avoid overlooking a flow pattern beyond the occlusion and to determine whether repair is possible.

Nonspastic hemifacial spasm confirmed by abnormal muscle responses.

BACKGROUND: Hemifacial spasm is usually diagnosed by inspection which mainly identifies involuntary movements of orbicularis oculi. Assessing abnormal muscle responses (AMR) is another diagnostic method. CASE DESCRIPTION: We report a case of left hemifacial spasm without detectable involuntary facial movements. The patient was a 48-year-old man with a long history of subjective left facial twitching. On magnetic resonance imaging (MRI), the left VIIth cranial nerve was compressed by the left anterior inferior cerebellar artery (AICA), which was in turn compressed by the left vertebral artery. We initially treated him with botulinum toxin. We were able to record AMR, and hemifacial spasm occurred after AMR stimulation, although no spasm was detectable by inspection. Subsequently, we performed microvascular decompression with transposition of the AICA that compressed the VIIth cranial nerve. His hemifacial spasm resolved by 5 weeks after surgery and was not induced by AMR stimulation. CONCLUSION: Hemifacial spasm can sometimes be diagnosed by detecting AMR rather than by visual inspection. We propose that such hemifacial spasm should be termed nonspastic hemifacial spasm.

KRIT1 mutations in three Japanese pedigrees with hereditary cavernous malformation.

Cerebral cavernous malformation is a neurovascular abnormality that can cause seizures, focal neurological deficits and intracerebral hemorrhage. Familial forms of this condition are characterized by de novo formation of multiple lesions and are autosomal-dominantly inherited via CCM1/KRIT1, CCM2/MGC4607 and CCM3/PDCD10 mutations. We identified three truncating mutations in KRIT1 from three Japanese families with CCMs: a novel frameshift mutation, a known frameshift mutation and a known splice-site mutation that had not been previously analyzed for aberrant splicing.

[Radiation-Induced Malignant Peripheral Nerve Sheath Tumor of the High Cervical Spine].

UNLABELLED: CASE: A 30-year-old woman presented with posterior cervical pain and left-sided omalgia. The patient had a history of non-Hodgkin's lymphoma for which she had received prophylactic whole-brain irradiation(including at the upper cervical level)17 years previously. A magnetic resonance imaging(MRI)scan obtained 1 month previously showed an intradural extramedullary mass lesion at the left C1/2 level. We initially considered the tumor to be a benign schwannoma, but the patient subsequently developed left hemiparesis and was consequently admitted 2 days after her first visit. A second MRI scan showed that the tumor had progressed markedly. Hence, the patient underwent emergency surgical excision of the tumor. However, the tumor could only be partially removed because it had strongly adhered to the ventral aspect of the spinal cord. The tumor was pathologically diagnosed as a malignant peripheral nerve sheath tumor(MPNST). The residual tumor was subjected to local irradiation and surgery, but the treatment was unsuccessful, and the patient died on the 91st day of her illness. Conclusion:We report a case of radiation-induced high cervical MPNST arising from a benign schwannoma. All 9 previously reported cases of radiation-induced spinal MPNST were reviewed. Intraspinal MPNST of the high cervical region are extremely rare and are associated with a very poor prognosis. The 5-year survival rate of such tumors is markedly worse than that of other types of MPNST, and no standard treatment has been established for this condition.

[Two Cases of Primary Intracranial Solitary Fibrous Tumor:Genetic Examination of NAB2-STAT6 Fusion and Its Association with Hemangiopericytoma].

Using whole exome and transcriptome sequencing, NAB2-STAT6 gene fusions have recently been identified in patients with solitary fibrous tumors (SFT). We report two cases of SFT in which NAB2-STAT6 fusions were identified. The patients were a 32-year-old man with a parasagittal tumor involving the superior sagittal sinus, and a 40-year-old man with a cerebellar convexity tumor partially involving the transverse sinus. Their tumors were gross totally resected and diagnosed to be SFT according to the following pathological findings:the tumors were composed of spindle cells with a patternless architecture, with prominent stromal collagen and staghorn vessels. An immunohistochemical study yielded positive results for CD34, CD99, and Bcl-2 and negative results for EMA, GFAP, and S100. The MIB-1 indexes were 13 and 7%, respectively. NAB2-STAT6 fusions were detected in both cases with a common fusion variant, NAB2ex6-STAT6ex16/17. We also identified NAB2-STAT6 fusions in two hemangiopericytomas diagnosed in the past with a common variant of NAB2ex6-STAT6ex16/17. These findings suggest that solitary fibrous tumor and hemangiopericytoma may be diagnosed based on the presence of NAB2-STAT6 fusion, and not classified separately because of the same genetic background.

Asymmetric epoxidation of allylic alcohols catalyzed by vanadium-binaphthylbishydroxamic Acid complex.

A vanadium-binaphthylbishydroxamic acid (BBHA) complex-catalyzed asymmetric epoxidation of allylic alcohols is described. The optically active binaphthyl-based ligands BBHA 2a and 2b were synthesized from (S)-1,1'-binaphthyl-2,2'-dicarboxylic acid and N-substituted-O-trimethylsilyl (TMS)-protected hydroxylamines via a one-pot, three-step procedure. The epoxidations of 2,3,3-trisubstituted allylic alcohols using the vanadium complex of 2a were easily performed in toluene with a TBHP water solution to afford (2R)-epoxy alcohols in good to excellent enantioselectivities.

[Clinical features of bilateral trigeminal neuralgia].

Among 238 patients with bilateral trigeminal neuralgia(TN)who visited our hospital between April 2007 and June 2014, 5(2%)were surgically treated by microvascular decompression(MVD). The initial symptom was on the right side in four and on both sides in one patient. Intervals between the initial and second onset on the other side(left)were two months, and four, six, and eight years. None of the patients showed involvement of the first branch of the trigeminal nerve. The patients with bilateral TN were younger than the 154 patients with unilateral TN who were treated surgically by MVD in this period(45 vs. 65 years), and the bilateral TN patients predominantly were women(4/5 vs. 99/154). In the surgical field, the trigeminal nerve and root entry zone were compressed more by veins in the bi lateral TN patients than in the unilateral TN(4/5 vs. 60/154, respectively)patients. We could not identify any differences in MRI CISS before versus after the onset of left trigeminal neuralgia, suggesting that compression is not the sole cause of the symptom.

[Trigeminal neuralgia caused by cerebellopontine angle lipoma:a case report and review of the literature].

A 59-year-old man presented with right trigeminal neuralgia of the second branch, which had been treated with carbamazepine. The pain could not be controlled adequately because of side effects. CT and MRI revealed a 2-cm lesion in the right cerebellopontine angle. Retrosigmoid lateral suboccipital craniectomy was performed, and a soft yellowish mass was found to be associated with the 5th, 7th, and 8th cranial nerves, anterior inferior cerebellar artery, and small vessels. The lipoma was partially resected from around the root entry zone(REZ)of the 5th nerve and small vessels were coagulated around the REZ. After surgery, there was no trigeminal neuralgia, but facial numbness and cerebellar signs were noted. Postoperative MRI showed decompression of the trigeminal nerve and venous infarction in the middle cerebellar peduncle. Reviewing similar cases, we found 19 lipoma patients presenting with trigeminal neuralgia. Symptoms of involvement of other cranial nerves were also present in 11 patients, and 14 were younger than 30 years old. Of 17 surgical cases, total resection was not attempted apart from one case. Although pain relief was achieved in all surgical cases, complications developed in 11. Surgery should be performed only in patients with disabling and uncontrolled symptoms.