This study reports on an extremely rare case of non-traumatic atlantoaxial rotatory fixation (AARF) in an adult. Although there are numerous reports on traumatic AARF in adults, those on non-traumatic AARFs are limited. We present the case of a 25-year-old woman who developed neck pain with a limited range of motion (ROM) that began upon waking without any particular inducement. Physical examination showed a characteristic torticollis neck posture called the cock robin position. The patient exhibited severely limited neck mobility with no neurological deficits. Computed tomography (CT) revealed that the atlas was rotated to the right in relation to the axis, resulting in a diagnosis of AARF Fielding type 1. Conservative treatment was adopted by immobilizing the cervical spine using a soft neck collar. Four weeks post-treatment, all symptoms improved with no adverse complications. We present an extremely rare case of non-traumatic AARF in an adult patient. As surgical treatment may be necessary if the diagnosis is delayed, appropriate diagnosis and treatment should be made in the early stages.

A 73-year-old man with chronic obstructive pulmonary disease received the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) mRNA vaccine. The following day, the patient developed a headache, followed by a tonic-clonic seizure and decreased consciousness. Magnetic resonance imaging of the head revealed no signs of stroke but multiple vasoconstrictions. Despite antiepileptic therapy, the seizure persisted, and the patient died 40 hours after vaccination. An autopsy revealed multiple brain ischemia without any vascular lesions, suggesting reversible cerebral vasoconstriction syndrome (RCVS). In this case, RCVS was diagnosed radiographically and pathologically. Our case suggests that RCVS could be a cause of headache and epilepsy following the SARS-CoV-2 mRNA vaccination.

A bony mallet thumb is an extremely rare injury. An 82-year-old man fell from a standing height and injured his right thumb. Imaging examinations revealed a rare intra-articular fracture at the dorsal side of the base of the distal phalanx of the thumb called the bony mallet thumb. Conservative treatment was adopted initially; however, surgery was deemed necessary because of the redislocation of the bone fragment. Thus, the Ishiguro extension block technique was used, and three months later, satisfactory thumb function was achieved. The Ishiguro technique is a relatively simple procedure often performed for bony mallet fingers. The current case indicated that it can also be used to treat cases of bony mallet thumbs successfully.

Central hypothyroidism and dyslipidemia are well-known adverse events (AEs) of bexarotene therapy. Although hypothyroidism is known to cause dyslipidemia, no study has examined the association between hypothyroidism and dyslipidemia in patients undergoing bexarotene therapy. The aim of this study is to examine this association. A retrospective observational study was performed among 294 patients who initiated bexarotene therapy in Japan (nation-wide postmarketing complete surveillance). Jonckheere-Terpstra (one sided) test was performed to evaluate the effect of the bexarotene dose on lipid metabolisms, and regression analyses were performed to evaluate associations of bexarotene dose, free thyroxine (FT4), body mass index (BMI), and lipid metabolisms. Most patients developed hypothyroidism. Two-third of patients showed FT4 values below the lower limit at 1 week. Triglycerides (TG) increased in a bexarotene dose-dependent manner, and grade ≥3 AEs on hypertriglyceridemia was observed in 39% of the patients. Additionally, one-third of grade ≥3 AEs on hypertriglyceridemia occurred within 1 week. The delta_FT4 (difference in FT4 from baseline) negatively correlated with TG increase at 1 week (p = 0.012) but not with low density lipoprotein cholesterol (LDL-C) increase at any week. Bexarotene-induced hypothyroidism is almost inevitable and occurred quickly. Bexarotene-induced hypertriglyceridemia showed positive bexarotene dose dependency and negative delta_FT4 dependency. Prophylactic and appropriate thyroid hormone compensation therapy and starting bexarotene at low doses with subsequent titration while managing dyslipidemia may have a beneficial effect for the successful continuation of bexarotene therapy without severe endocrine and metabolic AEs.

Abstract Background The prognosis of the walking ability of individuals with stroke affects the choice of rehabilitation program. Identifying patients who will need assistance with ambulation at discharge allows clinicians to deliver rehabilitation programs focused on educating caregivers and adjusting the patient’s environment to allow safe transfer within the home. The primary objective of this study was to develop and internally validate a prediction model of walking dependence for patients with stroke admitted to a rehabilitation facility. The secondary objective was to establish a prediction model of restricted walking speed. Methods This retrospective cohort study included 476 individuals with subacute stroke who were admitted to a rehabilitation facility. Model 1 was developed to predict the probability of walking dependence. Model 2 was developed to predict restricted walking speed. Walking dependence was defined as a functional independence measure walk score of 5 or less. Restricted walking speed was defined as the ability to walk at 0.93 m/s or less. Potential predictors, including age, leg strength of the affected side, sitting balance, cognitive function, and urinary function, were selected based on the literature and analyzed using logistic regression analysis. Models were internally validated using the bootstrap method. Model performance was assessed by discrimination (area under the receiver operating characteristic curve (AUC)) and calibration (Hosmer–Lemeshow (H–L) goodness-of-fit test and calibration plots). Results A total of 236 patients (49.6%) walked dependently at discharge. Of the 240 individuals who achieved walking independence, 121 (50.4%) had restricted walking speed. In model 1, older age, poor leg strength, sitting balance, cognitive function, and urinary incontinence were significantly associated with walking dependence at discharge. The AUCs of models 1 and 2 were 0.93 (95% confidence interval (CI) = .90–.95) and 0.69 (95%CI = .62–.76), respectively. Both models had good calibration confirmed by the H-L test. Conclusions The internally validated prediction model of walking dependence had good discrimination and calibration, while the prediction model of restricted walking speed had poor discrimination. The prediction model for walking dependence developed in this study may be useful for planning rehabilitation strategies and setting realistic goals for patients.

Posterior reversible encephalopathy syndrome (PRES) and cerebral infarction are both caused by hypertension, but they rarely occur together. If they do coexist, the selection of a management strategy is difficult because of their pathologic differences. Here, we present an uncommon case of brainstem and cerebellar PRES combined with acute lacunar infarction. For this patient, we used an aggressive blood pressure-lowering regimen during the acute phase of his condition. Once the cerebral edema caused by PRES began to improve, antiplatelet therapy was initiated. The treatment was ultimately successful, and the patient was discharged home. A return to work is now planned. Given the rarity of this combination of conditions and a lack of published evidence for management, our report will contribute to the literature concerning the treatment for this combination of conditions.

Precise immunolocalization of molecules in relation to ultrastructural features is challenging, especially when the target is small and not frequent enough to be included in tiny ultrathin sections randomly selected for electron microscopy (EM). Glucose transporter 1 (GLUT1) is in charge of transporting glucose across brain capillary endothelial cells (BCECs). Paraformaldehyde-fixed floating sections (50 μm thick) of mouse brain were immunolabeled with anti-GLUT1 antibody and visualized with fluoronanogold. Fluorescent images encompassing the entire hemisphere were tiled to enable selection of GLUT1-positive BCECs suitable for subsequent EM and landmark placement with laser microdissection to guide trimming. Sections were then fixed with glutaraldehyde, gold enhanced to intensify the labeling and fixed with osmium tetroxide to facilitate ultrastructural recognition. Even though a region that contained target BCECs was successfully trimmed in the resin block, it was only after observation of serial ultrathin sections that GLUT1 signals in coated vesicles on the same cross section corresponding to the cross section preidentified by confocal laser microscope. This is the first ultrastructural demonstration of GLUT1 molecules in coated vesicles, which may well explain its functional relevance to transport glucose across BCECs. Successful ultrastructural localization of molecules in relation to well-preserved target structure in native tissue samples, as achieved in this study, will pave the way to understand the functional relevance of molecules and their relation to ultrastructural details.

症例は79歳男性．主訴は脱肛．直腸指診，肛門鏡および大腸内視鏡検査で肛門7時方向に表面平滑な球状の柔らかい褐色の大きな腫瘤を認めた．MRI検査で内部壊死を認めない6cm大の線維性腫瘤と診断された．巨大な線維性肛門ポリープと術前診断し，腰椎麻酔下に切除した．腫瘤は6.0×3.8×3.5cmで，病理組織学的にはCD34陽性の間質細胞が豊富な線維性肛門ポリープと診断された．4cmを超える線維性肛門ポリープは稀であるため，報告例を集計し自験例を含めた7症例を比較検討した．大きな線維性肛門ポリープの多くは有症状で孤立性であり，長い病悩期間を有していた．本例のように褐色調で柔らかいポリープの報告例も認められた．