Internal Medicine
Online ISSN : 1349-7235
Print ISSN : 0918-2918
ISSN-L : 0918-2918
Advance online publication
Displaying 1-50 of 334 articles from this issue
  • Tomoyo Shimada, Ryota Kamo, Kensuke Daida, Kenya Nishioka, Nobutaka Ha ...
    Article ID: 8400-21
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    We experienced a young patient who presented with progressive parkinsonism and cerebellar ataxia. Brain magnetic resonance imaging revealed progressive brain calcification, expanding from the bilateral basal ganglia to the central pons, caused by a delayed reaction to the radiation therapy that she had received to treat craniopharyngioma 14 years earlier. Heterogeneous clinical symptoms due to radiation-induced brain calcification have been described, but parkinsonism has never been reported. While dopamine transporter-single photon emission computed tomography revealed only slight damage to the dopaminergic striatal pathway, the extension of calcification to the periventricular white matter was likely responsible for her parkinsonism.

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  • Takahiko Tsutsumi, Mika Ishibashi, Momoko Takemura, Shota Isashiki, Ry ...
    Article ID: 8726-21
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Objective Quantification of patient encounters during internal medicine residency training is challenging. At present, there are no established strategies for evaluating the whole inpatient experience in Japan. We hypothesized that the Diagnosis Procedure Combination (DPC) database, which is widely used in Japan, might be a useful tool for such an evaluation.

    Methods We analyzed DPC-based patient encounters of five senior residents with different types of training. One of the diseases on receipt computation data, including the four main diseases and at most eight comorbidities, was matched with each category in the Online system for Standardized Log of Evaluation and Registration of specialty training system (J-OSLER), and the match ratios were assessed. The accumulation of each disease classified into J-OSLER categories was also assessed. Monthly extra working hours and total patient-days per resident were evaluated using a Pearson correlation analysis.

    Results Two residents with two-year rotations in the general internal medicine department showed high numbers of patient encounters and the highest matching ratio with J-OSLER (approximately 60% with 4 major diseases, 91% with all diseases). There was a moderately positive correlation between the total patient-days and extra working hours in these residents, but no such correlation was noted in the rate of monthly patient encounters and extra working hours among residents as a whole.

    Conclusion The DPC-based quantification of patient encounters during residency training appears effective in evaluating the coverage of the current J-OSLER list. Owing to its wide availability and generalization, this matching method may be useful as a universal tool for assessing internal medicine programs.

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  • Masaoki Hidaka, Masaya Kumamoto, Tatsuya Suenaga, Ayaka Itsuji, Yuka K ...
    Article ID: 8910-21
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    A 30-year-old man admitted with renal dysfunction (serum creatinine, 8.19 mg/dL) was diagnosed with immunoglobulin A nephritis through a renal biopsy. He was treated with intravenous methylprednisolone pulse therapy and urgent hemodialysis, and eventually, he underwent maintenance hemodialysis. On day 108, he developed amnesia. Magnetic resonance imaging revealed bilateral basal ganglia lesions. Wernicke encephalopathy (WE) was diagnosed based on decreased serum thiamine concentration (12.8 μg/dL; reference range, 24-66 μg/dL). Thiamine replacement therapy was initiated, but the Wernicke-Korsakoff syndrome persisted. Careful monitoring of thiamine is required in patients undergoing dialysis. In addition, patients with WE may exhibit bilateral basal ganglia lesions.

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  • Ami Aoki, Akira Saito, Kenjiro Shima, Yosuke Kimura, Katsuaki Asakawa, ...
    Article ID: 9008-21
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    We herein report a 45-year-old-man with multiple foreign body granulomas in the lungs caused by polytetrafluoroethylene (PTFE). A mass in the right lower lobe of the lung and bilateral centrilobular lung nodules were found unexpectedly during the patient's visit to a hospital for a respiratory infection. The patient's occupation for 26 years involved spraying PTFE. A lung biopsy using bronchoscopy revealed granulomatous lesions and giant cells. The presence of fluorine in the granulomatous lesions was confirmed using an electron probe microanalyzer with wavelength dispersive spectrometer. Fluorine is a component of PTFE and is not found in normal lung tissue.

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  • Kana Takayama, Yoshinari Obata, Yumiko Maruo, Hiroki Yamaguchi, Motohi ...
    Article ID: 9179-21
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Metformin-associated lactic acidosis (MALA) is an extremely rare but life-threatening adverse effect of metformin treatment. The lifestyle changes associated with the coronavirus disease 2019 (COVID-19) pandemic may increase the potential risk of MALA development in patients with diabetes. We herein report a 64-year-old Japanese man taking a small dose of metformin who presented with MALA accompanied by hypoglycemia secondary to increased alcohol consumption triggered by lifestyle changes during the pandemic. Physicians should prescribe metformin judiciously to prevent MALA development and pay close attention to lifestyle changes in patients at risk for MALA during the COVID-19 pandemic.

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  • Hirohisa Fujikawa, Toshichika Mitsuyama, Daisuke Son, Masashi Izumiya, ...
    Article ID: 9268-21
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Objective Vaccination technique is a crucial skill for medical trainees to learn, especially in the current coronavirus disease 2019 pandemic. To this end, validated assessment tools are essential in teaching appropriate techniques. However, valid instruments for assessing vaccine administration skills have not yet been developed. We therefore explored the development and validation of an assessment tool for vaccination techniques based on expert consensus.

    Methods We implemented a modified Delphi process to develop a vaccination technique assessment tool. We then conducted a validation study to establish the reliability and validity of the tool.

    Results Two rounds of the modified Delphi process were performed to generate a 19-item, vaccination performance assessment checklist. In the validation study, the linear weighted kappa value for inter-rater reliability of the overall checklist score was 0.725. Spearman's correlation coefficient between the mean checklist score and the global rating was 0.98 (p <0.01).

    Conclusions This is a pioneering study examining the development and validation of an assessment tool for vaccine administration techniques. The tool will be widely used in vaccination-related education.

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  • Hiroki Ueno, Narumi Ohno, Takafumi Abe, Kazuki Kimoto, Chika Matsuoka, ...
    Article ID: 9296-21
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Central nervous system methotrexate-associated lymphoproliferative disorder (CNS-MTX-LPD) is rare, but its spontaneous regression has been observed in some patients after withdrawal of agents. We herein report three cases of primary CNS-MTX-LPD that received oral MTX for rheumatoid arthritis. Epstein-Barr virus and oligoclonal bands (OCBs) were positive, while proton magnetic resonance spectroscopy (1H-MRS) showed an elevated lipid peak and slightly elevated choline/N-acetylaspartate ratio in common. After MTX withdrawal, brain lesions showed spontaneous regression in all cases. Our patient's 1H-MRS findings and OCBs may reflect a non-monoclonal lymphoproliferative histology as benign-type lesions in CNS-MTX-LPD.

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  • Hiroaki Sakagami, Kanichi Otowa, Michiro Maruyama, Kazuo Usuda
    Article ID: 9322-22
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Seisuke Saito, Nobuhiro Ueno, Yuki Kamikokura, Yuya Sugiyama, Yu Kobay ...
    Article ID: 9590-22
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    A 24-year-old woman was admitted to our hospital due to abdominal pain and a high fever. She was diagnosed with ileocolonic Crohn's disease (CD), complicated with a gastro-colic fistula and splenomegaly. After initial treatment with an infliximab-biosimilar, all blood cell line counts markedly decreased. Three-dimensional reconstructed computed tomography revealed splenic vein narrowing. Thus, her pancytopenia was deemed to have likely been caused by hypersplenism. Surgery was performed, and clinical remission was maintained without pancytopenia. This is the first report of a CD patient with pancytopenia caused by hypersplenism that was triggered by gastro-colic fistula-associated splenic vein obstruction.

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  • Yasufumi Masaki, Yusuke Ueda, Hiroto Yanagisawa, Kotaro Arita, Tomoyuk ...
    Article ID: 9622-22
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    TAFRO syndrome was first described in 2010, standing for thrombocytopenia, anasarca, fever, reticulin fibrosis and organomegaly. Because the lymph node histopathology of TAFRO syndrome mimics idiopathic multicentric Castleman disease (iMCD), some researchers consider TAFRO syndrome to be a subtype of iMCD. However, the clinical features of TAFRO syndrome considerably differ from those of iMCD without TAFRO. The clinical features of patients with TAFRO syndrome with or without iMCD-histopathology are similar, and these patients require an accurate diagnosis and urgent treatment. Although a histological diagnosis, including a differential diagnosis, is important, lymph node involvement in patients with TAFRO syndrome is usually modest or sometimes absent. Furthermore, a bleeding tendency due to thrombocytopenia and severe anasarca hampers performing a biopsy. Nonetheless, patients with various other disorders may manifest TAFRO syndrome-like symptoms, making the differential diagnosis in borderline cases difficult. Therefore, the establishment of precise and specific biomarkers is important.

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  • Takahiro Kobayashi, Yosuke Ono, Naoya Fujita, Yuji Tanaka
    Article ID: 9766-22
    Published: 2022
    Advance online publication: May 21, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Naohiro Sekiguchi
    Article ID: 0041-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Kiyoshi Mori
    Article ID: 0044-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Koya Nagase, Yoshiro Fujita
    Article ID: 0047-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Yasuteru Inoue, Makoto Nakajima, Yuichiro Inatomi, Teruaki Masuda, Yoh ...
    Article ID: 8630-21
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    A 59-year-old man with progressive cognitive decline and mood disturbances was admitted to the hospital. Brain magnetic resonance imaging revealed marked white matter hyperintensity (WMH) and widespread lobar cerebral microbleeds. Because he had untreated hypertension, we started antihypertensive treatment and found a significantly improved cognitive function and WMH regression. We diagnosed him with cerebral amyloid angiopathy (CAA) based on the modified Boston Criteria with the rare apolipoprotein (ApoE) ε2/ε4 genotype.The mechanism underlying reversible leukoencephalopathy in CAA may be related to the loss of autoregulation of brain circulation: cerebrovascular Aβ deposits damaged the blood-brain barrier of the capillaries, which led to vasogenic edema induced by blood pressure surges.

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  • Ryuji Sakakibara, Yuki Sekiguchi, Jalesh N. Panicker, Noritoshi Sekido ...
    Article ID: 8724-21
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    We herein report a 73-year-old Japanese woman with possible multiple system atrophy-cerebellar form (MSA-C) who suffered from urinary retention (sacral autonomic disorder) for 12 years before exhibiting cerebellar ataxia. A peculiar combination of findings on urodynamics and sphincter electromyography (EMG), e.g. detrusor hyperactivity with impaired contraction (DHIC), detrusor-sphincter dyssynergia (DSD) and neurogenic sphincter EMG (upper and lower neuron-type autonomic dysfunction), seems to have been predictive of future development of MSA.

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  • Fumiya Kutsuna, Yoichi Morofuji, Eri Shiozaki, Daiki Uchida, Masahiro ...
    Article ID: 8906-21
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Bow hunter's stroke is a rare cause of vertebrobasilar infarction. There is no consensus regarding the optimal treatment. We herein report a case of bow hunter's stroke successfully treated by endovascular treatment. A 70-year-old man presented with central vertigo. Magnetic resonance imaging (MRI) showed posterior circulation infarcts. Dynamic angiography revealed thrombus formation and hypoperfusion of the right vertebral artery upon head rotation to the left. Endovascular parent artery occlusion of the right vertebral artery was performed, and there was no recurrence at follow-up MRI. Endovascular parent artery occlusion might be a useful treatment for bow hunter's stroke.

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  • Yusuke Yoshimura, Naoki Sawa, Shusaku Matsuoka, Daisuke Ikuma, Yuki Ob ...
    Article ID: 9185-21
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    We experienced a 77-year-old Japanese man who presented with nephrotic-range proteinuria 20 days after receiving ramucirumab treatment for metastatic sigmoid colon cancer. A kidney biopsy showed two characteristic histological findings. The first finding was podocyte injury with cellular crescent-like formation, although focal segmental glomerulosclerosis (FSGS) (collapsing variant) according to the Colombia classification may have been a more appropriate name for this injury, as hypertrophy and hyperplasia of epithelial cells, presumably resulting from podocyte injury, were seen between Bowman's capsule and the glomerular basement membrane (GBM); these changes appeared to be due to the collapse of the GBM rather than to GBM destruction with fibrinoid necrosis. The second finding was endotheliopathy characterized by prominent mesangial interposition via enlargement of the mesangial matrix with mesangiolysis. Proteinuria and renal dysfunction subsided after discontinuation of ramucirumab. Bevacizumab has been reported to induce glomerular microangiopathy with endothelial damage and swelling six months after treatment, but in this case, ramucirumab may have induced FSGS collapsing variant and glomerular microangiopathy with endotheliopathy via mesangial damage within 1 month. We believe that the damage to the glomerular podocyte and endothelial cells via mesangial damage secondary to ramucirumab in our patient was a different type of glomerular microangiopathy than the endothelial cell damage with enlargement of the subendothelial space caused by bevacizumab.

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  • Nobumasa Tamura, Shota Uchiyama, Saiko Nishioka, Kentaro Tamura, Masah ...
    Article ID: 9262-21
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Objective Coronavirus disease 2019 (COVID-19) has caused a collapse of the medical care system, with effective triage proving vital. The Kanagawa admission priority assessment score, version-1 (KAPAS-1) and version-2 (KAPAS-2), was developed to determine the need for hospitalization. Patients with a high KAPAS (≥ 5) are recommended for hospitalization. We retrospectively investigated the correlation between the KAPAS and oxygen requirement during hospitalization.

    Methods We collected the clinical data of COVID-19 patients admitted between February 5 and December 6, 2020. Patients were divided into two groups: those who required oxygen therapy during hospitalization (OXY) and those who did not (NOXY). We assessed the correlations between the groups and KAPAS-1 and KAPAS-2.

    Results Overall, 117 COVID-19 patients were analyzed, including 20 OXY and 97 NOXY and 54 high KAPAS-1 and 63 high KAPAS-2. The median KAPAS-1 and KAPAS-2 were significantly higher in OXY than in NOXY (6.5 vs. 3, and 9 vs. 4, respectively). The areas under the receiver operating characteristic curves of KAPAS-1 and KAPAS-2 for oxygen requirement were 0.777 and 0.825, respectively, and the maximum values of Youden's index were 4 and 6, respectively. The proportions of high KAPAS-1 and high KAPAS-2 were significantly higher in OXY than in NOXY (90.0% vs. 37.1%, and 90.0% vs. 46.4%, respectively).

    Conclusion The KAPAS was significantly correlated with oxygen requirement. Furthermore, the KAPAS may be useful for deciding which patients are most likely to require hospitalization and for selecting non-hospitalized patients who should be carefully monitored.

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  • Tsugumi Hosoe, Kenzo Uzu, Kiyohiro Hyogo, Ryosuke Takahashi, Sho Tamai ...
    Article ID: 9305-21
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Swallow or deglutition syncope is an unusual disorder. We herein report an 80-year-old man with paroxysmal atrial tachycardia induced by swallowing, causing syncope. Initially, we suspected a digestive disorder and found no significant findings. Finally, a swallowing test with monitoring of the heart rate and blood pressure helped in the diagnosis. The patient was treated with antiarrhythmic drugs and catheter ablation. The mechanism underlying swallowing-induced tachycardia presumably involves mechanical stimulation of the esophagus and autonomic nervous system effects. However, few cases have been reported, and the exact mechanism remains unclear.

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  • Bonpei Takase, Katsumi Hayashi, Satoko Takei, Tetsuya Hisada, Nobuyuki ...
    Article ID: 9318-21
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Coronavirus disease 2019 (COVID-19) has become a pandemic, and vaccines remain the only effective tools available for ending it. However, their side effects, such as syncope, which mimics sudden cardiac death, are serious concerns. We herein report 6 cases of delayed vasovagal syncope and presyncope (VVR) caused by COVID-19 vaccination among 25,530 COVID-19 patients. The prevalence of delayed VVR due to COVID-19 vaccination was 0.026%. In addition, no delayed VVR was found among 17,386 patients who received the influenza vaccine. Delayed VVR is likely to be overlooked if medical staff are not aware of this symptom. This report provides significant information regarding effects of COVID-19 vaccination.

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  • Yuichiro Iwamoto, Shigehito Mori, Fuminori Tatsumi, Toshitomo Sugisaki ...
    Article ID: 9365-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Background Pituitary inflammation due to IgG4-related disease is a rare condition and is sometimes accompanied by central diabetes insipidus. Central diabetes insipidus produces a strong thirst sensation, which may be difficult to distinguish when complicated by salivary insufficiency.

    Case presentation A 45-year-old man was admitted to our department for a thorough examination of his thirst and polyuria. He had suddenly developed these symptoms more than one year earlier and visited an oral surgeon. Swelling of the left submandibular gland, right parotid gland, and cervical lymph nodes had been observed. Since his IgG4 level was relatively high at 792 mg/dL and a lip biopsy showed high plasmacytoid infiltration around the gland ducts, he had been diagnosed with IgG4-related disease. He had started taking 0.4 mg/kg/day of prednisolone, and his chief complaint temporarily improved. However, since the symptom recurred, he was referred to our institution. After admission, to examine the cause of his thirst and polyuria, we performed a water restriction test, vasopressin loading test, hypertonic saline loading test and pituitary magnetic resonance imaging. Based on the findings, we diagnosed him with central diabetes insipidus due to IgG4-related hypophysitis. We increased the dose of prednisolone to 0.6 mg/kg/day and started 10 μg/day of intranasal desmopressin. His symptoms were subsequently alleviated, and his serum IgG4 level finally normalized.

    Conclusion We should remember that IgG4-related disease can be accompanied by hypophysitis and that central diabetes insipidus is brought about by IgG4-related hypophysitis. This case report should remind physicians of the fact that pituitary inflammation due to IgG4-related disease is very rare and can be masked by symptoms due to salivary gland inflammation, which can lead to pitfalls in the diagnosis in clinical practice.

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  • Teruyuki Ishikura, Tatsusada Okuno, Toshiyuki Takahashi, Hideki Mochiz ...
    Article ID: 9446-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Soichi Ohta, Takeo Matsuyoshi, Hitoshi Kaneko, Daiyu Kosen, Hiroaki Su ...
    Article ID: 9472-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Regarding extracorporeal membrane oxygenation (ECMO) support against hemorrhagic conditions, there seems to be a dilemma when deciding between maintaining the circuit patency by systemic anticoagulation and increasing the risk of bleeding. We herein report two cases of diffuse alveolar hemorrhaging (DAH) caused by myeloperoxidase (MPO) anti-neutrophil cytoplasmic autoantibody-associated vasculitis (AAV) successfully treated with venovenous (VV)-ECMO support, both initially started without systemic anticoagulation. Under anticoagulation-free ECMO management, we should consider the shortcomings of frequent circuit exchange and hemorrhagic diathesis related to circuit-induced disseminated intravascular coagulation (DIC).

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  • Itaru Goto, Ryuji Okamoto, Yu Sato, Akihiro Tsuji, Takashi Tanigawa, K ...
    Article ID: 9516-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Ebstein's anomaly is an uncommon congenital disorder affecting the tricuspid valve. We herein report a 38-year-old woman who experienced consciousness and sensory disturbance during treatment for heart failure caused by Ebstein's anomaly. Urgent magnetic resonance imaging and cerebral angiography demonstrated acute cerebral infarction and internal carotid artery obstruction with the development of collateral arteries. We diagnosed her with multiple cerebral infarctions due to moyamoya disease. Ebstein's anomaly concomitant with moyamoya disease is extremely rare. However, we should consider the possibility of this rare but important concurrence when treating patients with heart failure due to Ebstein's anomaly to avoid excessive diuresis and vasodilation and irreversible brain injury.

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  • Satoshi Akao, Akitsu Higuchi, Kei Akao, Ryo Rokutanda
    Article ID: 9517-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Hirokazu Kawai, Toshifumi Sato, Masaaki Natsui, Kotaro Watanabe, Ryosu ...
    Article ID: 9526-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    A 76-year-old man presented with liver dysfunction and intrahepatic bile duct dilatation. Imaging studies showed two large stones that had become impacted in the common hepatic duct, which was fused with the gallbladder. The patient was diagnosed with Mirizzi syndrome type IV. Hepaticojejunostomy and stone removal failed due to dense gallbladder adhesions involving the right hepatic artery. The bile flow was temporarily restored; however, the patient experienced cholangitis 16 months later. The stones were extracted via peroral single-operator cholangioscopy (SOC)-guided electrohydraulic lithotripsy. This is the first case in which stones were completely removed by SOC-guided treatment in a patient with Mirizzi syndrome type IV.

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  • Takashi Oyama, Kazuki Taoka, Akira Chiba, Kensuke Matsuda, Hiroaki Mak ...
    Article ID: 9545-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Bing-Neel syndrome (BNS) is a rare central nervous system manifestation of lymphoplasmacytic lymphoma/Waldenström macroglobulinemia (LPL/WM). We herein report a 62-year-old man with LPL/WM after multiple chemotherapies. He had weakness of lower extremities and elevated serum IgM levels. A bone marrow examination showed lymphoplasmacytic cells infiltration. Contrast-enhanced magnetic resonance imaging suggested enhancing lesions in the cauda equina roots. He was diagnosed with BNS and started on treatment with tirabrutinib 480 mg daily. Within three months, he showed clinical and radiologic improvement. Tirabrutinib may have utility as an effective treatment for BNS.

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  • Hajime Yasuda, Taku Hatano, Tadahiro Honda, Miyuki Tsutsui, Nobutaka H ...
    Article ID: 9577-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Vitamin B6 (VB6) is essential to heme synthesis, and its deficiency can lead to anemia. VB6 deficiency anemia is typically microcytic, hypochromic, and sideroblastic. VB6 deficiency is a well-recognized complication of levodopa/carbidopa therapy, as metabolism of levodopa to dopamine is VB6-dependent, and carbidopa irreversibly forms bonds and deactivates VB6. We herein report a 75-year-old man with advanced Parkinson's disease who developed severe VB6 deficiency anemia due to levodopa/carbidopa intestinal gel therapy. His anemia was promptly resolved with simple oral supplementation of pyridoxal phosphate hydrate. VB6 deficiency anemia can mimic myelodysplastic syndrome and thus is an important differential diagnosis for patients administered levodopa/carbidopa.

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  • Toshihide Izumida, Teruhiko Imamura
    Article ID: 9642-22
    Published: 2022
    Advance online publication: May 14, 2022
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  • Hangyul Kim, Hye-Ree Kim, Min Gyu Kang, Kyehwan Kim, Jin-sin Koh, Jeon ...
    Article ID: 9652-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Intermittent left main coronary artery ostium obstruction (LMOO) caused by native aortic valve thrombus (NAVT) is an extremely rare condition. It may therefore be challenging to identify the cause using only coronary angiography, even though the clinical presentation and electrocardiography (ECG) strongly suggest myocardial infarction. We herein report a 53-year-old man with NAVT complicating intermittent occlusion of left main disease in preexisting coronary artery stenosis.

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  • Yoshiaki Kinoshita, Takuto Miyamura, Hisako Kushima, Hiroshi Ishii
    Article ID: 9656-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Kosaku Komiya, Marcelo Takahiro Mitui, Toru Watanabe, Masaru Nasu, Kaz ...
    Article ID: 9747-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    A man in his 70s visited our department for dyspnea with pulmonary infiltrate that was unresolved by antibiotics. He had been taking Sansoninto for five years and doubled its dose a month ago. After discontinuing Sansoninto without any additional medications, his symptoms gradually disappeared, and pulmonary infiltration improved. Drug lymphocyte stimulation tests showed a positive result for Sansoninto. We diagnosed this patient with Sansoninto-induced lung injury. Sansoninto is a combination drug that consists of sansonin, bukuryo, senkyo, chimo, and kanzo. This paper reports the first case of Sansoninto-induced lung injury and discusses the mechanism considering its components.

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  • Tomoaki Yamasaki, Yuhei Sakata, Takehisa Suekane, Hiroko Nebiki
    Article ID: 9817-22
    Published: 2022
    Advance online publication: May 14, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Hideki Uedono, Akihiro Tsuda, Noriko Ueno, Yuka Natsuki, Rino Nakaya, ...
    Article ID: 8820-21
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Systemic lupus erythematosus (SLE) is a chronic autoimmune inflammatory disease. Lupus nephritis (LN) is a major risk factor for mortality in SLE, and glomerular "full-house" immunofluorescence staining is a well-known characteristic of LN. However, some cases of non-lupus glomerulonephritis can also present with a "full-house" immunofluorescence pattern. We recently encountered a patient with full-house nephropathy (FHN) during adalimumab administration for Crohn's disease. IgA nephropathy or idiopathic FHN was diagnosed, and treatment with steroids was started, after which there was improvement in proteinuria. The prognosis of FHN has been reported to be poor; therefore, aggressive treatment is required for such patients.

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  • Yuta Nanjo, Tomoko Okuma, Yumi Kuroda, Eri Hayakawa, Kohei Shibayama, ...
    Article ID: 9065-21
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Objective Based on the increasing incidence of smell and taste dysfunction among coronavirus disease 2019 (COVID-19) patients, such issues have been considered an early symptom of infection. However, few studies have investigated the type of taste components that are most frequently affected in COVID-19 patients. This study investigated the difference in frequencies of the types of taste component disorders among hospitalized COVID-19 patients.

    Methods In this retrospective, single-center, observational study, patients' background characteristics, clinical course, laboratory and radiological findings, and details on taste and/or smell disorders were collected and analyzed from medical records.

    Patients A total of 227 COVID-19 patients were enrolled, among whom 92 (40.5%) complained of taste disorders.

    Results Multiple types of taste disorders (hypogeusia/ageusia and hypersensitivity, or hypersensitivity and changing tastes) were reported in 10 patients. In particular, 23 patients reported hypersensitivity to at least 1 type of taste, and 2 patients complained of a bitter taste on consuming sweet foods. Impairment of all taste components was found in 48 patients (52.2%). The most frequent taste disorder was salty taste disorder (81 patients, 89.0%). Hypersensitivity to salty taste was most frequently observed (19 patients, 20.9%).

    Conclusion Patients with COVID-19 develop multiple types of taste disorders, among which salty taste disorder was the most frequent, with many patients developing hypersensitivity to salty taste. As smell and taste are subjective senses, further studies with the combined use of objective examinations will be required to confirm the findings.

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  • Myung-Won Lee, Sang-Hoon Yeon, Hyewon Ryu, Ik-Chan Song, Hyo-Jin Lee, ...
    Article ID: 9124-21
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Objective We retrospectively analyzed the prevalence and clinical features of splenic infarctions in patients with Philadelphia chromosome-negative myeloproliferative neoplasms (Ph- MPNs).

    Patients Patients diagnosed with essential thrombocythemia (ET), polycythemia vera (PV), prefibrotic/early primary myelofibrosis (pre-PMF), or PMF from January 1996 to October 2020 in Chungnam National University Hospital, Daejeon, Korea, were reviewed.

    Results A total of 347 patients (143 ET, 129 PV, 44 pre-PMF, and 31 PMF patients; 201 men and 146 women) with a median age of 64 (range 15-91) years old were followed up for a median of 4.7 (range 0.1-26.5) years. Fifteen (4.3%) patients exhibited splenic infarctions at the diagnosis. These were most common in PMF patients (12.9%), followed by pre-PMF (9.1%) and PV (5.4%) patients. Multifocal infarcts (60.0%) were most common, followed by solitary (33.3%) and extensive infarcts (6.7%). The cumulative incidence of thrombosis in patients with splenic infarctions tended to be higher than in those lacking infarctions (10-year incidence 46.7% vs. 21.0% in PV; p=0.215; 33.3% vs. 17.9% in pre-PMF; p=0.473) patients, but statistical significance was lacking. Palpable splenomegaly (HR 14.89; 95% CI 4.00-55.35; p<0.001) was the only independent risk factor for splenic infarction. During follow-up, 5 (1.4%) patients developed splenic infarctions. Conservative treatment adequately controlled the symptoms; no serious complications were noted in any patient.

    Conclusion Splenic infarctions occurred most frequently in patients with PMF; it was rare in patients with ET. The clinical courses were generally mild.

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  • Kousuke Baba, Tokiko Fukuda, Mitsuru Furuta, Satoru Tada, Atsuko Imai, ...
    Article ID: 9221-21
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Phosphoglycerate kinase (PGK) deficiency is an X-linked disorder characterized by a combination of hemolytic anemia, myopathy, and brain involvement. We herein report a Japanese man who had several episodes of rhabdomyolysis but was training strenuously to be a professional boxer. Mild hemolytic anemia was noted. The enzymatic activity of PGK was significantly reduced, and a novel missense mutation, p.S62N, was identified in the PGK1 gene. A literature review revealed only one case with a mixed hemolytic and myopathic phenotype like ours. This mild phenotype indicates the complex pathophysiology of PGK deficiency and suggests the benefits of dietary control and exercise.

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  • Kazumasa Soma, Dai Nakamatsu, Tsutomu Nishida, Yutaro Minoura, Miyoung ...
    Article ID: 9361-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Objective Treatment for uncomplicated diverticulitis (UD) is not well established. We evaluated the strategy of reviewing intravenous antibiotics for hospitalized Japanese patients with UD.

    Methods Treatment was based on the physician's choice until August 2018; the indications for hospitalization and treatment have been standardized since September 2018. In this study, we monitored the use of intravenous antibiotics administered to patients hospitalized for UD and then reviewed the need for them on hospital day 3. We compared patients' length of antibiotic use, hospital stay, health care cost, and complications via the review strategy from September 2018 to December 2020 and via the previous physicians' choice strategy from January 2016 to August 2018.

    Results Two hundred and forty-seven patients were admitted to our hospital because of acute colonic diverticulitis from January 2016 to December 2020. After excluding complicated cases, 106 individuals were enrolled during the period of physician's choice; 87 were enrolled when treatment review was employed. There were no significant differences in age, sex, inflammation site, or severity during the first hospital visit. The median duration of antibiotic use was significantly reduced from 5 to 4 days (P=0.0075), with no marked increase in rates of transfer to surgery, mortality, or readmission due to recurrence. A more significant proportion of patients completed 3-day antibiotic treatment with the review strategy than with the physician's choice strategy (6.6% vs. 25.3%, P=0.0004). However, the length of hospital stay and total medical costs did not decrease.

    Conclusions The strategy of reviewing treatment on day 3 after hospitalization for UD safety reduced the duration of antibiotic use, but the hospital stay and health care costs did not decrease.

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  • Ruanxian Dai, Qiang Meng
    Article ID: 9376-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Posterior reversible encephalopathy syndrome is an uncommon syndrome in exertional rhabdomyolysis. We herein report a case of rhabdomyolysis and acute kidney injury after intense exercise. The patient also had generalized hypertension, headache, and painless complete loss of vision in both eyes, which was consistent with the symptoms of posterior reversible encephalopathy syndrome detected by magnetic resonance imaging of the brain. The patient fully recovered through active fluid replacement, diuresis, blood pressure control, alkalization of urine and blood purification, and supportive measures. This case report describes an infrequent neurological complication of exertional rhabdomyolysis. Understanding the range of complications is critical for improving patient outcomes.

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  • Hajime Nakamura, Kohichi Takada, Makoto Emori, Naotaka Hayasaka, Shint ...
    Article ID: 9423-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    We herein report a rare case of unresectable liposarcoma that showed a complete response to eribulin. Furthermore, a low expression of phosphorylated AKT (p-AKT) on an immunohistological evaluation was observed. This result is consistent with our previous preclinical study that demonstrated the significance of p-AKT signaling for eribulin resistance in multiple subtypes of soft tissue sarcoma (STS) cells. This case highlights the potential benefits of eribulin as well as the mechanism underlying resistance to eribulin in patients with unresectable or metastatic STS, especially liposarcoma.

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  • Hiroyuki Hokama, Yuki Sakamoto, Toshiyuki Hayashi, Seira Hatake, Mizuh ...
    Article ID: 9439-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    The pathophysiology of unilateral cortical fluid-attenuated inversion recovery (FLAIR)-hyperintense lesions in anti-myelin oligodendrocyte glycoprotein (MOG)-associated encephalitis with seizures (FLAMES) is unclear. A 26-year-old man was referred because of a seizure. FLAIR showed an increased signal intensity and swelling of the right frontal cortex. His symptoms and imaging abnormalities were improved after intravenous methylprednisolone therapy. MOG antibody was detected both in serum and cerebrospinal fluid (CSF). Therefore, the patient was diagnosed with FLAMES. Myelin basic protein (MBP) was elevated in CSF. The high MBP value in the CSF in the present case suggested that demyelination as well as inflammation can occur in some FLAMES patients.

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  • Sho Matsushita, Kenei Sada, Atsushi Manabe, Eriko Kashihara, Shunsuke ...
    Article ID: 9487-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    We encountered a case of refractory adult-onset Still's disease (AOSD) with two relapses. Prednisolone and methotrexate were begun as induction therapy, resulting in the patient's first relapse during tapering of prednisolone. After the introduction of tocilizumab, she achieved remission. However, she experienced a second relapse following prednisolone tapering. While lactate dehydrogenase (LDH) levels and white blood cell (WBC) counts increased in both relapses, interleukin-6 suppression resulted in stable C-reactive protein and ferritin levels in the second relapse. A comparison of the two relapses indicated that increases in both WBC counts and LDH levels can aid in the diagnosis of AOSD relapse.

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  • Morihisa Hirota, Keigo Murakami, Akinobu Koiwai, Keita Kawamura, Yuki ...
    Article ID: 9565-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    The histopathological findings of immune checkpoint inhibitor (ICI)-induced pancreatitis have rarely been reported. A 56-year-old man with squamous cell carcinoma of the lung with bone metastasis was being treated with pembrolizumab, an anti-programed cell death protein-1 antibody. After 13 doses, he was referred to our department due to pancreatitis. Despite characteristic symptoms of acute pancreatitis, imaging findings were similar to those of autoimmune pancreatitis. However, a histological examination showed neutrophil-based inflammatory cell infiltration and acinar-ductal metaplasia. Immunostaining showed CD8-positive T lymphocyte infiltration. This case revealed the characteristic histopathology of pembrolizumab-induced pancreatitis, which was previously poorly understood.

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  • Yuki Takigawa, Hiromi Watanabe, Daiki Kagawa, Ken Sato
    Article ID: 9657-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Sho Sasaki, Kazuo Mizumoto, Akira Sasaki, Taro Takami
    Article ID: 9682-22
    Published: 2022
    Advance online publication: May 07, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION
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  • Ryo Terayama, Takahiro Ishikawa, Kazuki Ishiwata, Ai Sato, Takuya Mina ...
    Article ID: 8326-21
    Published: 2022
    Advance online publication: April 30, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Cushing's disease causes numerous metabolic disorders, cognitive decline, and sarcopenia, leading to deterioration of the general health in older individuals. Cushing's disease can be treated with transsphenoidal surgery, but thus far, surgery has often been avoided in older patients. We herein report an older woman with Cushing's disease whose cognitive impairment and sarcopenia improved after transsphenoidal surgery. Although cognitive impairment and sarcopenia in most older patients show resistance to treatment, our case indicates that normalization of the cortisol level by transsphenoidal surgery can be effective in improving the cognitive impairment and muscle mass loss caused by Cushing's disease.

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  • Takashi Owaki, Satoru Hashimoto, Hajime Umezu, Shuji Terai
    Article ID: 8728-21
    Published: 2022
    Advance online publication: April 30, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    The most common sites of breast cancer metastasis include the lymph nodes, bones, lungs, liver, and brain. Gastrointestinal tract metastasis is rarely seen, and hypopharyngeal metastasis is extremely rare. We herein report a case of late distant recurrence of breast cancer and synchronous metastasis to the hypopharynx, stomach, ileum, bones, and lymph nodes almost 24 years after surgery. To our knowledge, this is the first case of synchronous metastasis to the hypopharynx, gastrointestinal tract, and other organs, especially after a long interval following primary mastectomy.

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  • Akio Nakata, Hideyuki Takamatsu, Yuya Eguchi, Toshihide Izumida, Isao ...
    Article ID: 8936-21
    Published: 2022
    Advance online publication: April 30, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    Primary cardiac lymphomas (PCLs) are extremely rare tumors with a poor prognosis. They usually involve the pericardium and the right side of the heart. PCLs arising from the left side of the heart are relatively rare, with bilateral cases being even rarer. We herein report a case of PCL arising from both the right and left sides of the heart in a 65-year-old man. Multiple imaging modalities clearly demonstrated the tumors at the initial evaluation. The pathological diagnosis was diffuse large B-cell lymphoma.

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  • Shuhei Okuyama, Hidetoshi Satomi, Ryuto Ishikawa, Tsutomu Shishido, Ke ...
    Article ID: 9300-21
    Published: 2022
    Advance online publication: April 30, 2022
    JOURNAL OPEN ACCESS ADVANCE PUBLICATION

    A 58-year-old man presented with painful edema of the extremities, and a diagnosis of eosinophilic fasciitis (EF) was confirmed. He also met the criteria for hypereosinophilic syndrome (HES), but there were no findings suggestive of malignancies or hematologic neoplasms despite a close examination. He was started on steroid therapy but subsequently developed severe liver dysfunction, hemophagocytic lymphohistiocytosis, hepatosplenomegaly, and renal involvement. The diagnosis of peripheral T-cell lymphoma, not otherwise specified was finally established by a bone marrow reexamination and liver biopsy. In cases of eosinophilia, EF, and/or HES, it is important to suspect an intrinsic abnormality, including potential T-cell lymphoma.

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