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1.
ObjectivesEosinophilic otitis media (EOM) is an intractable otitis media mostly associated with bronchial asthma. Dupilumab, an anti-interleukin (IL)-4 receptor (R)α, is effective and has been approved for use in patients with moderate to severe bronchial asthma, atopic dermatitis and chronic rhinosinusitis with nasal polyposis, whose diseases are not controlled by previous treatments including other molecular targeted drugs. We aimed to assess efficacy of dupilumab in three EOM patients with associated bronchial asthma, who were poor responders to previous topical and systemic corticosteroid therapy and molecular targeted therapies.Patients and methodsThree patients with severe, refractory EOM (two with a granulation type) associated with bronchial asthma received dupilumab as add-on therapy for at least 6 months. The efficacy of dupilumab therapy was evaluated using severity scores, symptom scores, hearing acuities, temporal bone computed tomography (CT) scores, and surrogate markers before and after therapy.ResultsSeverity scores in all patients were dramatically reduced to 2 points or less (full score: 16 points) after initiation of therapy. Air conduction hearing levels were improved in all patients. Temporal bone CT scores in two patients were reduced, and serum IgE levels in all three patients also decreased following therapy.ConclusionWe provide the first report that add-on dupilumab therapy was effective in patients with severe, refractory EOM who did not respond to the treatments including other molecular targeted therapy. Patients with severe middle ear mucosal change may benefit particularly from dupilumab therapy. 相似文献
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Nobusuke Kimura Yukitoshi Takahashi Hideo Shigematsu Katsumi Imai Hiroko Ikeda Hideyuki Ootani Rumiko Takayama Yukiko Mogami Noriko Kimura Koichi Baba Kazumi Matsuda Takayasu Tottori Naotaka Usui Satohiko Kondou Yushi Inoue 《Brain & development》2019,41(1):77-84
Objective
The purpose of this study was to identify the risk factors of cognitive impairment in pediatric epilepsy patients with focal cortical dysplasia (FCD).Methods
77 patients with histopathologically confirmed FCD were studied. The statistical relationship between cognition levels and clinical factors at presurgical evaluation was analyzed. Cognitive function was evaluated by development quotient or intelligence quotient (DQ-IQ).Results
Ages at seizure onset were younger than 15?years (mean?±?SD; 5.0?±?4.2?years). Mean disease duration was 14.5?±?8.5?years. Mean age at pre-surgical DQ-IQ evaluation was 34.8?±?10.7?years. Mean DQ-IQ was 60.5?±?20.5, and 41 of 77 (53.2%) patients had mental retardation (DQ-IQ?<?70). Younger seizure onset and seizure clustering were significantly associated with lower DQ-IQ (p?<?0.001). A multiple regression study identified higher seizure frequency pattern, a history of epileptic spasm and status epilepticus as aggravating factors of DQ-IQ decline (R2?=?0.63, p?<?0.001). On the other hand, the risk was decreased in patients with habitual focal aware seizure and transient seizure-free periods up to 6?months in the course of epilepsy. FCD location (FCD site, extent of radiological lesion and laterality) and histopathology of FCD did not affect DQ-IQ.Conclusions
Our study suggests that seizure characteristics including higher seizure frequency pattern, a history of epileptic spasm, status epilepticus, seizure clustering and early onset of seizure are risk factors of cognitive impairment in FCD patients. 相似文献3.
4.
Shingo Hashimoto Masaki Katsurada Rie Muramatsu Kumiko Asai Kenichiro Tanaka Kensuke Hayashi Yoshiaki Kibe Koichiro Nakajima Yukiko Hattori Hiromitsu Iwata Jun-etsu Mizoe Hiroyuki Ogino Yuta Shibamoto 《Practical radiation oncology》2019,9(2):e149-e155
Purpose
Suppression of respiratory movement of the liver would be desirable for high-precision radiation therapy for liver tumors. We aimed to investigate the effect of our original device-free compressed shell fixation method and breathing instruction on suppression of respiratory movement. The characteristics of liver motion based on the movement of a fiducial marker were also analyzed.Methods and Materials
First, respiratory amplitudes of the liver with the device-free compressed shell were analyzed from the data of 146 patients. The effect of this shell fixing method on liver movement was evaluated. Second, as another cohort study with 166 patients, interfractional internal motion of the liver for patients fixed in the shell was calculated using the fiducial marker coordinate data of images for position setting before daily irradiation. Third, in another 12 patients, intrafractional internal motion was calculated from the fiducial marker coordinate data using x-ray images before and after irradiation.Results
The median respiratory movement without the shell, after fixing with the shell, and after instructing on the breathing method with the shell was 14.2 (interquartile range, 10.7-19.8), 11.5 (8.6-17.5), and 10.4 mm (7.3-15.8), respectively. Systematic and random errors of interfractional internal motion were all ≤2 mm in the left-right and anteroposterior directions and 3.7 and 3.0 mm, respectively, in the craniocaudal direction. Systematic and random errors of intrafractional internal motion were all ≤1.3 mm in the left-right and anteroposterior directions and 0.8 and 2.4 mm, respectively, in the craniocaudal direction.Conclusions
The device-free compressed shell fixation method was effective in suppressing the respiratory movement of the liver. Irradiation position matching using the fiducial marker can correct the interfractional internal motion on each day, which would contribute to the reduction of the margin to be given around the target. 相似文献5.
Takako Fujita Yukiko Ihara Hitomi Hayashi Atsushi Ishii Hiroshi Ideguchi Takahito Inoue Taichi Imaizumi Toshiyuki Yamamoto Shinichi Hirose 《Congenital anomalies》2020,60(6):189-193
Coffin-Siris syndrome (CSS) is a congenital anomaly syndrome characterized by developmental delay, coarse facial features, and hypoplasia of the fifth digit's nail or phalanges. Herein, we report a case of the 8-year-old female patient who showed developmental delay associated with dysplasia in the macular and large toe area. Comprehensive genomic analysis showed no possible candidate variants, but the subsequent genomic copy number analysis revealed a novel exonic deletion in the coding region of AT-rich interactive domain-containing protein 1B (ARID1B), a gene responsible for CSS. Genomic copy number analysis can aid in diagnosing CSS by confirming undiagnosed exonic deletions in ARID1B. Furthermore, this is the first report of CSS associated with bilateral macular dysplasia. 相似文献
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Naoko Shoji Keiji Tanese Ayano Sasaki Taishi Horiuchi Yuji Utsuno Koichi Fukuda Yukiko Hoshino Shinichi Noda Hirofumi Minami Wataru Asakura Amenamevir Review Team 《The Journal of dermatology》2020,47(7):683-688
In July 2017, Japan’s Ministry of Health, Labor and Welfare issued a marketing authorization valid throughout Japan for N-(2,6-dimethylphenyl)-N-(2-{[4-(1,2,4-oxadiazol-3-yl)phenyl]amino}-2-oxoethyl)-1,1-dioxothiane-4-carboxamide (amenamevir) for the first time worldwide. The decision was based on the favorable opinion of the Pharmaceuticals and Medical Device Agency (PMDA) recommending a marketing authorization of amenamevir for treatment of herpes zoster (HZ). Amenamevir has a different action mechanism from previously approved synthetic nucleoside compounds for the treatment of HZ including acyclovir, valacyclovir and famciclovir. The usual adult dose is 400 mg amenamevir p.o. once daily for 7 days. The benefit is its ability to cure HZ as well as preventing postherpetic neuralgia. The most common side-effects are increase of urine N-acetyl-β-D-glucosaminidase and α1-microglobulin levels. However, based on the detailed evaluation of the submitted clinical studies, there seems to be no serious safety concerns about amenamevir regarding the kidney of both renally normal and impaired patients. The objective of this article is to summarize the scientific review of the application. The detailed scientific assessment report and product information, including the summary of product characteristics, are available on the PMDA website ( www.pmda.go.jp/PmdaSearch/iyakuSearch/ ). 相似文献
9.
Yutaka Fujioka Nobuhiro Hata Ryusuke Hatae Satoshi O. Suzuki Yuhei Sangatsuda Yukiko Nakahara Masahiro Mizoguchi Koji Iihara 《Neuropathology》2020,40(1):99-103
Diffuse midline glioma, H3 K27M mutant arises from midline structures of the central nervous system and predominately affects pediatric patients. However, this disease entity was only recently established, and the clinical phenotypic spectrum remains largely unclear. We herein report a rare case of diffuse midline glioma, H3 K27M mutant with an unusual distribution in an elderly woman who presented with a diffuse glioma that invaded both sides of the thalami, and left hippocampus and frontoparietal lobes, thus mimicking a hemispheric malignant glioma. A biopsy of the lobular lesion led to a molecular diagnostic confirmation of diffuse midline glioma, H3 K27M mutant. The patient received concurrent bevacizumab and temozolomide therapy with radiation therapy and survived for 30 months. This case highlights the possibility that a glioma with cerebral hemispheric spread in an elderly patient may harbor the H3 K27M mutation. 相似文献
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