Double Skull Sign After Cranioplasty: A Case Report.

The double skull sign (DSS) is a unique image on the outside of the brain that looks like two skulls. Whereas congenital and acquired types of DSS have been reported, the etiology of both of them is calcified hematomas. We encountered a case of a 46-year-old woman with a history of subarachnoid hemorrhage followed by cranioplasty at 43 years old. She developed right hemiparalysis and motor aphasia suddenly. Brain computed tomography and magnetic resonance imaging revealed not only cerebral infarction but also DSS incidentally. After detailed analysis, we concluded that the DSS in this case was not due to calcification of the hematoma but was related to the cranioplasty. In this report, we present an interesting case and discuss etiologies of the development of DSS after cranioplasty.

Characteristics of Screw Perforation and Screw Loosening in Atlantoaxial Transarticular Fixation Using a Preoperative Computed Tomography-Based Navigation System.

BACKGROUND: Atlantoaxial transarticular fixation, also called the Magerl technique, is said to be the most robust biomechanical method of fixation of the atlantoaxial vertebrae. However, the procedure carries a risk of spinal cord and vertebral artery injury during the insertion process, especially in patients with a high-riding vertebral artery. In this study, a computed tomography (CT)-based navigation system was used for preoperative planning and insertion. This investigation sought to determine the rate and direction of screw perforation as well as the incidence of screw loosening in computer-assisted atlantoaxial transarticular fixation. METHODS: Sixty patients (31 men and 29 women; mean ± SD age: 65.3 ± 19.6 years) who received atlantoaxial transarticular screw insertion with preoperative CT navigation were analyzed. We investigated screw position and loosening by CT at the final follow-up. RESULTS: Of the 108 screws inserted, the rate of Grade 2 or higher perforation was 4.6% (5/108). Nine of 81 (11.1%) screws inserted into the 44 patients who were followed for at least 6 months showed loosening. Logistic regression analysis revealed that unilateral insertion (odds ratio: 8.50, 95% confidence interval: 1.53-47.2, P = 0.014) was significantly associated with the incidence of screw loosening. CONCLUSIONS: The screw perforation rate of Grade 2 or higher in computer-assisted atlantoaxial transarticular screw fixation was 4.6%, with comparable frequencies of perforation direction. Unilateral insertion was a significant independent factor associated with screw loosening, which occurred in 11.1% of insertions. CLINICAL RELEVANCE: Spine surgeons should follow up with patients with caution because screws with unilateral insertion are prone to loosening.

Treatment of Subcortical Aphasia Due to Putaminal Hemorrhage With the Japanese Version of Melodic Intonation Therapy (MIT-J).

Melodic intonation therapy (MIT) is one of the rehabilitation methods for patients with non-fluent or dysfluent aphasia, mainly caused by stroke or brain injury. Although MIT is conducted in various languages, reports on the Japanese version of MIT (MIT-J) are limited. In this report, we describe a case about the efficacy of MIT-J in the subacute phase after stroke on subcortical aphasia. Our case was a 60-year-old right-handed woman who suffered from left putaminal hemorrhage. She was treated with acute therapy, including medications and rehabilitation, but non-fluent aphasia was preserved. Regardless of general speech therapies, her aphasia was not improved. In the subacute phase, we started MIT-J (protocol: 20 minutes per day, five days per week for two weeks). The effect of MIT-J was remarkable and in particular, speech intelligibility was improved. It is required to accumulate more cases to reveal the effect of MIT-J.

Painless Thyroiditis by Withania somnifera (Ashwagandha).

The Withania somnifera, also called Ashwagandha, is available everywhere in the world. We present a rare case of thyrotoxicosis following Ashwagandha administration, specifically painless thyroiditis (PT) in this report. The patient was a 47-year-old previously healthy Japanese man, who started taking Ashwagandha two months before his first visit to our hospital. He visited our hospital for typical thyrotoxicosis symptoms like a sense of fatigue, fever at night, and weight loss followed by diarrhea and headache. Blood tests disclosed thyrotoxicosis. Thyroid ultrasonography showed internal echo heterogeneity and no increase in blood flow. Thyroid scintigraphy revealed a deficiency in thyroid uptake. Based on these findings, he was diagnosed as PT. After stopping the administration of Ashwagandha, both his symptoms and serum thyroid markers were improved. This report may spark important debate about whether ashwagandha is safe among healthy people, especially in thyroid toxicity.

Efficacy of Trihexyphenidyl on Apraxia of Eyelid Opening in Parkinsonism: A Case Report.

Apraxia of eyelid opening (AEO) is occasionally seen in Parkinson's disease (PD) or related diseases. However, many clinicians have trouble with the management of AEO by Parkinsonism. In this report, we describe a case of AEO in Parkinsonism improved by trihexyphenidyl (THP). The patient was a 64-year-old woman, who was previously healthy but developed bradykinesia. She was clinically diagnosed as PD due to an L-dopa challenge test, but no other detailed tests were performed. She started antiparkinsonian medications and her symptoms were improved at an early phase. However, her motor symptoms were gradually exacerbated over time, and antiparkinsonian medications were dosed up. At 69 years old, blepharospasm and AEO developed. Although other antiparkinsonian medications did not improve her AEO, THP cured AEO dramatically at 73 years old. In this report, we discuss a mechanism of AEO by Parkinsonism and the pathway of THP for the improvement of AEO.

Miller-Fisher Syndrome Following Influenza A Infection.

Miller-Fisher syndrome (MFS), characterized by ophthalmoplegia, ataxia, and areflexia, is a Guillain-Barré syndrome (GBS) variant. It is well-known that the causative antibody for MFS is anti-GQ1b antibody. This report describes a rare case of MFS with not only anti-GQ1b antibodies but also anti-GT1a antibodies following Influenza A infection. The patient, a 47-year-old woman, contracted Influenza A three weeks before admission. She complained of double vision followed by areflexia, ataxia in the four extremities, and complete gaze palsy. She was treated with intravenous methylprednisolone pulse and intravenous immunoglobulin therapies. Her neurological symptoms were recovered after these immunotherapies.

[A Case of Lymphoepithelial Cyst of the Pancreas for Which Malignancy Could Not Be Ruled Out Difficult Preoperative Diagnosis].

Lymphoepithelial cyst(LEC)of the pancreas is a relatively rare benign cystic disease of the pancreas. In this report, we describe a case of LEC in which a malignant tumor could not be ruled out by preoperative diagnosis and surgery was performed. The patient was a 72-year-old man. A simple CT scan of the chest and abdomen performed as a follow-up for another disease incidentally revealed a mass in the pancreatic tail. Enhanced CT of the abdomen showed a tumor approximately 3 cm in size at the pancreatic tail with no contrast effect. MRCP showed moderate signal on T2WI, high signal on T1WI, and high signal on T2WI on some cysts inside the pancreas. PET-CT showed slight uptake of FDG. Both tumor markers CEA and CA19-9 were normal. Therefore, malignant disease such as pancreatic IPMC could not be ruled out, and laparoscopic distal pancreatectomy plus splenectomy was performed. The pathology results showed a diagnosis of pancreatic lymphoepithelial cyst with slight differentiation into sebaceous gland.

Hashimoto Encephalopathy and Thyroid Storm by Diabetic Ketoacidosis and Acute Pancreatitis: A Case Report.

Hashimoto encephalopathy (HE) is a rare condition related to autoimmune origin characterized by high titers of antithyroid antibodies. Steroids are effective for treatment of HE, suggesting the autoimmunity as an underlying mechanism. In addition, anti-NH2-terminal of α-enolase antibodies (anti-NAE antibodies) is useful for diagnosis of HE. This report describes a 69-year-old woman developing both HE and thyroid storm (TS), following diabetic ketoacidosis (DKA) and acute pancreatitis. She had a history of Basedow's disease and uncontrolled type 2 diabetes mellitus, and her serum hemoglobin A1c was 10%. She complained of nausea and visited our hospital. She was diagnosed with DKA and acute pancreatitis. After admission, she went into cardiopulmonary arrest and she was diagnosed with TS after resuscitation. In addition, blood test collected during acute phase of TS revealed positive for not only anti-thyroid peroxidase (TPO) antibodies, thyroid stimulating hormone receptor antibodies and thyroid stimulating antibodies, but also anti-NAE antibodies. She was treated with intravenous steroids, potassium iodide and thiamazole under respirator and recovered sufficiently to do daily activities of life. We should keep in mind that there might be cases of HE in cases of TS presenting with central nervous system symptoms.

Dural Arteriovenous Fistula in Neuro-Behçet's Disease: Association or Chance?

Behçet's disease (BD) is a multisystemic vasculitis disorder. Neuro-Behçet's disease (NBD) is a set of neurologic symptoms imputable to an underlying Behçet vasculitis. Among the wide range of vascular abnormalities secondary to BD, a dural arteriovenous fistula (dAVF) is not classically described. Whether a dAVF is associated with BD or dAVF is a chance occurrence is still a matter of debate. Herein, we describe an NBD case of a 48-year-old male, presenting with headache and fever, where a dAVF was seen on imaging. He was treated with prednisolone and colchicine, followed by the surgical resection for dAVF. Then, we discuss the possible association between BD and dAVF based on the latest literature.

Lateral Medullary Infarction With Atonic Bladder and Lateropulsion.

Lateral medullary infarction (LMI), or Wallenberg syndrome, can develop various symptoms, but it is rare that ipsilateral axial lateropulsion (or body lateropulsion, BL) or atonic bladder (AB) are caused by LMI. This report describes a case of LMI with both BL and AB. A 77-year-old man, with a history of hypertension and diabetes, developed acute left BL and anuresis. A neurological exam showed right gaze nystagmus, slight dysarthria and dysphagia, right dysesthesia in the trunk, and ataxia in the left limbs and trunk. Horner's syndrome and paralysis were unremarkable. Brain magnetic resonance imaging revealed hyperintensity in the lateral medulla oblongata. Cystometry revealed AB, although the patient had the urge to urinate. Owing to acute therapy, although trunk ataxia was presented for several months, BL and anuresis were recovered on day 15 and day 35, respectively. Here, we describe the potential mechanisms of BL and AB caused by LMI.

Pure Motor Isolated Finger Palsy by Cerebral Infarction: Tips for Diagnosis by Neurological Examination.

There are various causes of unilateral finger palsy. Its potential etiologies include peripheral neuropathy, carpal tunnel syndrome, and nerve root disorder due to myelopathy. In addition to them, similar paralysis has been reported in localized lesions of the cerebrum, classically referred to as pseudoperipheral palsy. In this report, we describe a case of an 80-year-old man who developed sudden clumsiness of the right fingers. Neurological examination showed muscle weakness mainly in the 1st and 2nd fingers (Medical Research Council grade 1-4) and normal reflexes in the extremities. The affected muscles were innervated by the median nerve, ulnar nerve, and radial nerve, and their nerve root levels ranged from C6 to T1. All the Phalen's, Tinel's, and flick signs were negative. Diffusion-weighted brain magnetic resonance imaging showed hyperintensity limited in the precentral knob on the left precentral gyrus. The etiology was diagnosed as cardiogenic embolism due to atrial fibrillation. In this report, we provide key findings for diagnosing pure motor isolated finger palsy by cerebral infarction through neurological examination.

Normal-Pressure Hydrocephalus-Like Appearance in Myotonic Dystrophy Type 1.

Myotonic dystrophy type 1 (DM1) is one of the monogenic neurological diseases that neurologists most often experience. DM1 can develop several symptoms, including muscle weakness, gait disturbance, urinary incontinence, and cognitive decline. Other hand, normal pressure hydrocephalus (NPH) is more frequent in the elderly population and is characterized by a triad of symptoms, gait disturbance, urinary urge incontinence, and cognitive decline. Therefore, some symptoms overlap between DM1 and NPH. In this report, we described a case of DM1 that presented with a triad of NPH, and NPH-like changes in brain images. A 54-year-old man with DM1 visited our hospital for rehabilitation. He had a history of dyslipidemia, diabetes, and cataracts. He developed muscle weakness, blepharoptosis, and dysarthria at 43 years. Neuro-exam revealed percussion and grip myotonia, distal muscle weakness and atrophy, broad-based gait, and urinary incontinence. The mini-mental state examination score was 18. Brain magnetic resonance imaging revealed enlarged lateral and third ventricles and Evans index was 0.38 (NPH criterion; >0.3), which was mimicking for NPH. Tap test (TT) was evaluated twice. First TT improved clinical symptoms slightly, but second was unremarkable. Based on the second TT result, we could not diagnose with NPH and could prevent unnecessary surgical shunting. Brain imaging of DM1 can show an NPH-like appearance in patients older than 50. Although TT is the gold standard for diagnosing NPH, its sensitivity and specificity vary among reports. TT results should be interpreted with caution before performing a surgical shunt. If necessary, multiple TTs should be considered in DM1 patients.

Clinical performance and shape analysis of trifocal intraocular lenses via scanning electron microscopy.

BACKGROUND: To evaluate visual performance after implantation of the TFNT (Acrysof Panoptix, Alcon, Fort Worth, Texas, USA) and CNWT (Clareon Panoptix, Alcon, Fort Worth, Texas, USA) intraocular lens (IOL), and compare the lens shape observed by scanning electron microscope (SEM). METHODS: Eighteen patients (18 eyes) received implantation of the CNWT and Twenty patients (20 eyes) received implantation of the TFNT. Exclusion criteria were previous ocular surgeries, ocular pathologies, or corneal abnormalities. Intervention or Observational Procedure(s): Postoperative examination at 1 months including manifest refraction; evaluation of refractive error, distance-corrected visual acuity (DCVA) at 5 m, 1 m, 70 cm, 50 cm, 40 cm, and 30 cm, slit-lamp examination; defocus curve testing; contrast sensitivity (CS) was performed. The lens shape of the TFNT and the CNWT was examined under SEM. RESULTS: Mean spherical equivalent was 0.11 ± 0.41 D (CNWT group) and 0.12 ± 0.34 D (TFNT group) 1 month postoperation. DCVA and defocus curve showed no significant difference between the two groups. CS was significantly higher in CNWT group than TFNT group at spatial frequencies of 6 cycles per degree (cpd). Observation of the IOL with a scanning electron microscope (SEM) revealed that CNWT group had improved diffraction structure and edge processing accuracy compared to TFNT group. CONCLUSION: There was no significant difference between the two groups in the defocus curve and visual acuity at all distances. CS was better in the CNWT group than in the TFNT group. IOL surface features may affect CS.

A Case of Autoimmune Polyendocrine Syndrome Type 3B and Peripheral Neuropathy Due to Thiamine Deficiency.

Autoimmune polyendocrine syndrome (APS) type 3B is characterized by presence of autoimmune thyroid disease, chronic atrophic gastritis and pernicious anemia. In this report, we present a rare case of APS type 3B with neuropathy by thiamine deficiency. A 65-year-old man had a history with hypothyroidism, gastritis, gastrectomy for gastric cancer and subacute combined degeneration of the spinal cord. Patient developed polyneuropathy with not mecobalamin but thiamine deficiency. Serum anti-thyroglobin (TG), anti-thyroid peroxidase (TPO), and anti-gastric parietal cell antibodies were positive. He was treated with thiamine supplementation and improved muscle weakness, sensory impairment and gait disturbance. Classically, it is reported gastric cancer related to hypothyroidism. Additionally, thiamine deficiency can be caused by gastrectomy. Here, his thiamine deficiency was related to APS type 3B, leading to polyneuropathy.

Is it safe to control the car pedal with the lower limb of the unaffected side in patients with stroke?

OBJECTIVES: Few studies have examined motor function in determining the suitability of patients with stroke to resume driving a car. Patients with hemiplegia usually control car pedals with the unaffected lower limb. However, motor control on the unaffected side is also impaired in patients with stroke. This study aimed to clarify the neurophysiological characteristics of pedal switching control during emergency braking in patients with hemiplegia. METHODS: The study participants consisted of 10 drivers with left hemiplegia and 10 age-matched healthy drivers. An experimental pedal was used to measure muscle activity and kinematic data during braking, triggered by the light from a light-emitting diode placed in front of the drivers. RESULTS: The patient group took the same reaction time as the healthy group. However, from the visual stimulus to the release of the accelerator pedal, the patient group had higher muscle activity in the tibialis anterior and rectus femoris and had faster angular velocities of hip and knee flexion than the healthy group. In addition, the patient group had higher co-contraction activities between flexors and extensors. From the accelerator pedal release to brake contact, the patient group had slower angular velocities of hip adduction, internal rotation, ankle dorsiflexion, internal return, and internal rotation than the healthy group. CONCLUSIONS: Patients with hemiplegia exhibited poor control of pedal switching using their unaffected side throughout the pedal-switching task. These results indicate that the safety related to car-pedal control should be carefully evaluated while deciding whether a patient can resume driving a car after a stroke.

Three Types of Demyelination, Perivenous, Confluent, and Perineuronal Nets-Rich in a COVID-19 Patient With Meningoencephalomyelitis.

Neurologic symptoms are common in COVID-19, and a variety of neuropathological changes have been reported. One of the important neuropathological findings is demyelination. However, the underlying pathogenesis of demyelination remained poorly understood. We witnessed a case of COVID-19 with distinct types of demyelination in the cerebrum, medulla oblongata, and spinal canal, who died of sepsis. The postmortem examination showed the solitary massive demyelination in the medulla oblongata. The massive lesion was filled with components of perineuronal nets. In the spinal canal, confluent demyelination in bilateral lateral and dorsal funiculi was detected over the entire length from C1 to S5, which was maximum at the level of cervical spinal canal stenosis. Demyelination in the cerebrum was mainly perivenular, and augmented in the area of lacunar infarcts and dilated perivascular spaces. Considering the distribution patterns of the following three types of demyelination, the traces of viral spreading could be highlighted. Discontinuous perivenous demyelination in the cerebrum showed the result of hematogenous spreading. Longitudinal confluent demyelination of the spinal cord should be the picturesque of the trace of axonal spreading. The distribution of demyelination was possibly modified by the underlying diseases, diabetes mellitus, hypertension, and spinal canal stenosis.

Case report: Clinically mild encephalitis/encephalopathy with a reversible splenial lesion: an autopsy case.

Clinically mild encephalitis/encephalopathy with a reversible splenial lesion is a clinicoradiological syndrome characterized by transient neuropsychiatric symptoms and hyperintensity of the splenium of the corpus callosum on diffusion-weighted MRI. Although intramyelinic edema and inflammatory cell infiltration can be predicted by MRI, the pathology of the splenium of the corpus callosum remains unknown. We encountered a case of clinically mild encephalitis/encephalopathy with a reversible splenial lesion and hypoglycemia in a patient who died of sepsis, and an autopsy was performed. The postmortem pathological findings included intramyelinic edema, myelin pallor, loss of fibrous astrocytes, microglial reactions, and minimal lymphocytic infiltration in the parenchyma. Based on these findings, transient demyelination following cytotoxic edema in the splenium of corpus callosum was strongly considered a pathogenesis of "clinically mild encephalitis/encephalopathy with a reversible splenial lesion" associated with hypoglycemia, and it could be generalized for the disease associated with the other causes. As cytotoxic edema could be the central pathology of the disease, the recently proposed term cytotoxic lesions of the corpus callosum may be applicable to this syndrome.

[Peritoneal Recurrence of Gastric Cancer Treated with Chemoradiotherapy-A Case Report].

A 76-year-old male patient underwent a distal gastrectomy for advanced gastric cancer. As the postoperative serum CA19-9 level was elevated, chemotherapy was initiated. Computed tomography(CT)detected a solitary peritoneal recurrence in the left subhepatic space 17 months later. Consequently, chemoradiotherapy(CRT)at a total dose of 60 Gy, combined with S-1 therapy, was administered for local tumor control. After CRT, CT scans revealed a remarkable reduction in the peritoneal recurrence. Presently, 8 months after CRT, the patient remains alive with no indications of regrowth. CRT could prove efficacious as a treatment for gastric cancer patients with localized peritoneal recurrences.

[A Case of Pathologic Complete Response Achieved with Preoperative Pembrolizumab Therapy for Transverse Colon Cancer].

A 77-year-old female patient presented with a medical history of 4 cancerous lesions, each with a surgical history. She was referred to our hospital due to anemia. Upon examination, she was diagnosed with transverse colon cancer. Duodenal invasion was suspected, which made performing R0 surgery difficult; therefore, the NAC approach was chosen. Three courses of CAPOX were administered, resulting in tumor obstruction, leading to the formation of an ileum stoma. MSI testing revealed MSI-H, and pembrolizumab treatment was initiated. CT scans showed tumor shrinkage, and PET scans indicated no accumulation, resulting in a cCR. Colon resection including the lesion suspected of stenosis was performed with a strong desire for stoma closure and the determination of potential curative resection. Additionally, a partial resection of the duodenum was performed. Pathological examination did not reveal any evident tumor cells, leading to the determination for a pCR. The patient has been under postoperative surveillance for 1 year without any recurrence.