Mesial Frontal Lobe Infarction Presenting as Pisa Syndrome.

Pisa syndrome is usually seen in patients with Alzheimer's disease treated with a cholinesterase inhibitor, dementia with Lewy bodies, Parkinson's disease, or atypical parkinsonism including multiple system atrophy. An 86-year-old woman presented with an acute onset of lateral flexion of her trunk to the left side, i.e., Pisa syndrome. She also showed left hemiparesis predominantly in her lower extremity. Her diffusion-weighted magnetic resonance images showed acute infarction in the right premotor area and supplementary motor area. Clopidogrel (75 mg daily) was prescribed. After two weeks from the onset of symptoms, her Pisa syndrome improved. The pathophysiology of Pisa syndrome has not yet been fully understood, but different mechanisms have been assumed. In this patient, it is possible that the infarction in her unilateral frontal lobe impaired the information processing from the temporoparietal cortex to the frontal lobe, including the premotor area and supplementary motor area for anticipatory postural control.

Perinatal outcome of pregnancy after adenomyomectomy: summary of 10 cases with a brief literature review.

Objectives: The purpose of this study was the perinatal outcomes of patients who became pregnant after adenomyomectomy.Study design: The retrospective cohort study was performed involving pregnant women with a history of adenomyomectomy between 1 January 2011 and 31 December 2018. At 24-26 weeks, the patients were admitted even without symptoms or signs. When regular uterine contractions were observed, tocolysis was performed.Results: Ten patients were included. Elective and emergent cesarean section (CS) was performed in seven and three patients, respectively. Emergent CS was performed due to onset of labor (tocolytic failure) at 28, 24, and 32 weeks. Although no patients suffered uterine rupture, myometrial thinning was observed at the site corresponding to that of adenomyomectomy in three patients. Of these three patients, two required emergent CS due to tocolytic failure with cervical length (CL) shortening. In contrast, CLs were stable in the other seven patients with elective CS.Conclusions: Three patients after adenomyomectomy showed preterm delivery, and three had a very thin uterus to the extent that the fetus could be observed through the uterine wall. A short CL should be paid special attention in pregnant women with a history of adenomyomectomy.

Multiple cerebral infarction diagnosed as Eosinophilic Granulomatosis with Polyangiitis by autopsy.

BACKGROUND: Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare systemic vasculitis of unknown cause involving the brain and accompanied by prominent eosinophilia. Intracardiac thrombosis is a major cardiac complication of EGPA that may cause thromboembolism. CASE PRESENTATION: A 53-year-old man presenting with abulia (consciousness disturbance) and left upper limb paralysis was admitted to our hospital. His case was complicated by penetrating branches, small vessel infarcts, and endocardial thrombosis in the right and left ventricle. Cardiomyopathy was also observed. Sixteen days after admission, the patient died from intracranial hemorrhage. Brain autopsy revealed two major findings: 1) large hemorrhagic infarction caused by cardiac embolism; and 2) granuloma and eosinophil infiltration. Vasculitis was accompanied by eosinophil infiltration in the cortical blood vessels and granuloma. CONCLUSIONS: In this case study, we report autopsy findings of brain infarction in a patient with EGPA and endocardial thrombosis. The brain infarction was caused by the cardiac embolisms and vasculitis.

Specific mechanisms of subarachnoid hemorrhage accompanied by ischemic stroke in essential thrombocythemia: two case reports and a literature review.

BACKGROUND: JAK2 V617F mutation increases the risk of thrombosis, and both ischemic and hemorrhagic strokes can occur in essential thrombocythemia (ET). The mechanisms underlying ischemic stroke in ET are diverse, and hemorrhagic stroke has rarely been reported in ET. METHODS: Among 627 stroke patients, those identified as having ET were investigated retrospectively. A comprehensive systemic literature search of the PubMed database was also conducted. RESULTS: Two cases were extracted with the diagnosis of ET who developed SAH and then ischemic stroke. In Case 1, a 47-year-old woman developed SAH in the left high convexity. Eleven hours later, acute cerebellar infarction suddenly developed due to right vertebral artery dissection. In Case 2, a 70-year-old woman developed SAH in the right high convexity. Magnetic resonance angiography showed multifocal stenotic changes in intracranial arteries. Three days later, she developed acute brain infarcts in the right middle cerebral artery territory. Eight weeks later, multifocal stenotic lesions improved. The literature review revealed 5 patients with hemorrhagic stroke and 40 patients with ischemic stroke associated with ET. Age at onset varied, female gender predominated, and the frequency of JAK2 V617F mutation was high. Atherosclerotic vascular risk factors were more common in ischemic stroke, but not in hemorrhagic stroke. CONCLUSIONS: The current study describes rare cases of SAH accompanied by ischemic stroke secondary to ET along with a review of the current literature, implying specific mechanisms for cerebral artery disorders associated with JAK2 V617F mutation.

Pontine hemorrhage accompanied by neuromyelitis optica spectrum disorder.

Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune disorder caused by antibody to aquaporin-4 (AQP4). NMOSD can infrequently present as a complication of posterior reversible encephalopathy syndrome (PRES). Moreover, few cases of NMOSD patients with brain hemorrhage have been reported. We report a rare case of PRES together with NMOSD recurrence, subsequent to pontine hemorrhage after intravenous methylprednisolone (IVMP) therapy. A 51-year-old Japanese woman, with a history of hypertension and dyslipidemia, and recurrent episodes of left visual acuity disorder related to AQP4-positive NMOSD, developed blindness in the left eye. Brain MRI showed a hyperintense lesion in pons. She was initially diagnosed with recurrence of NMOSD and 1000 mg of IVMP was administered for 3 days. After the 3rd course of IVMP, she developed left-sided sensory disturbance, and blood pressure was increased to 202/127 mmHg. Brain computed tomography (CT) showed pontine hemorrhage, and she was referred to our hospital again. We diagnosed PRES associated with NMOSD recurrence, along with development of pontine hemorrhage induced by the increase in blood pressure resulting from IVMP. The patient was treated with nicardipine to strictly control blood pressure, and tranexamic acid and glycerol for pontine hemorrhage and PRES. We also extended IVMP for 5 consecutive days in total, followed by plasmapheresis. After therapy, blindness in the left eye improved to light perception. Collectively, anti-AQP4 antibody could induce PRES together with recurrent NMOSD, and pontine hemorrhage could thus be induced by blood pressure increases resulting from IVMP.

Temporal Profile of CT and T2*-Weighted Gradient-Echo MRI in a Patient with Unilateral Thalamostriate Vein Thrombosis.

Deep cerebral venous system thrombosis (DCVST) is an uncommon variety of thrombosis that accounts for 11% of cases of cerebral venous thrombosis. Thalamostriate vein (TSV) thrombosis is further rare among patients with DCVST. Although patients with cerebral venous thrombosis commonly have characteristic neurological deficits including headache, deterioration of consciousness, and seizures, patients with DCVST do not necessarily show such symptoms. Therefore, diagnose of DCVST is sometimes difficult. Here we report a case of TSV thrombosis with a unilateral basal ganglion lesion presenting with right-sided hemiparesis. A 61-year-old Japanese female was referred to our hospital. On neurological examination, she had no headache but presented with right facial paresis with dysarthria. Her right hemiparesis was present in the upper and lower extremities. We repeatedly performed brain computed tomography (CT) and T2*-weighted conventional gradient-echo (GRE) magnetic resonance imaging, and conclusively diagnosed as left TSV thrombosis. We firstly report a case of unilateral DCVST associated with TSV thrombosis in which a temporal profile of CT and T2*-weighted GRE images was obtained. Although DCVST is a rare clinical entity, physicians should be aware that repeated radiological observations can be useful for the diagnosis and early medical treatment for DCVST.