論文 - 橋口　明典

Granulomatous interstitial nephritis with CTLA-4 haploinsufficiency: a case report

Kohatsu K., Suzuki T., Takimoto M., Matsui K., Hashiguchi A., Koike J., Shirai S.

BMC Nephrology （BMC Nephrology）  23 （ 1 ） 367 2022年12月

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　概要を見る

Background: Cytotoxic T lymphocyte antigen-4 (CTLA-4) is an essential inhibitory regulator of immune activation. CTLA-4 haploinsufficiency is known to be associated with dysregulation of FOXP3+ regulatory T cells, hyperactivation of effector T cells, and lymphocytic infiltration of multiple organs. However, there have only been a few reports of renal involvement with CTLA-4. Herein, we present a case of acute granulomatous tubulointerstitial nephritis (TIN) in a patient with CTLA-4 haploinsufficiency. Case presentation: A 44-year-old man presented with a 3-week history of fever and malaise, and subsequently developed acute kidney injury (AKI) a few days after treatment with levofloxacin (LVFX). A kidney biopsy and immunohistochemical staining revealed granulomatous TIN with dominantly infiltrating CD4+ T cells. General symptoms and renal impairment showed improvement after discontinuation of LVFX and initiation of oral steroids. However, they worsened following steroid tapering. Further, a colon biopsy analysis showed similar findings to the renal tissue analysis. We suspected that granulomatous TIN was possibly associated with CTLA-4 haploinsufficiency. Therefore, the patient was transferred to another hospital for further treatment of CTLA-4 haploinsufficiency using immunosuppressive agents. Conclusions: There have been few reports regarding renal involvement of CTLA-4 haploinsufficiency. In the present case, granulomatous TIN could have arisen due to instability of immune regulatory functions, such as CTLA-4 haploinsufficiency, and treatment with LVFX could have triggered immunologic activation and severe inflammation as well as renal dysfunction.

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肺炎球菌性菌血症で発見されたC3 nephritic factor陽性のC3腎炎/MPGNの7歳女児

森下 俊真, 遠藤 翔太, 武政 洋一, 大貫 裕太, 櫻谷 浩志, 村上 仁彦, 藤永 周一郎, 橋口 明典

日本小児腎臓病学会雑誌 （(一社)日本小児腎臓病学会）  35 （ 2 ） 175 - 176 2022年11月

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寛解後もC3低値が持続し追跡腎生検でC3NeF陽性C3腎炎と診断した1例

荒尾 正人, 飛田和 えりか, 吉村 萌, 秋岡 祐子, 橋口 明典

日本小児腎臓病学会雑誌 （(一社)日本小児腎臓病学会）  35 （ 2 ） 175 - 175 2022年11月

共著,  ISSN  0915-2245

尿細管・間質病変主体のANCA関連血管炎の一例

西岡 謙, 山口 慎太郎, 安田 格, 吉本 憲史, 田島 敬也, 林 香, 吉野 純, 橋口 明典, 神田 武志, 伊藤 裕

日本腎臓学会誌 （(一社)日本腎臓学会）  64 （ 6-E ） 549 - 549 2022年10月

共著,  ISSN  0385-2385

間質にIgM-λ陽性の沈着物を認めたprimary renal lymphomaの一例

川口 隆久, 唐澤 隆明, 橋口 明典, 大嶋 洋佑, 渡辺 雄祐, 熊谷 聡佑, 有馬 功一郎, 安藤 孝

日本腎臓学会誌 （(一社)日本腎臓学会）  64 （ 6-E ） 550 - 550 2022年10月

共著,  ISSN  0385-2385

妊娠末期に発症した膜性腎症の1例

伊藤 智章, 中村 健吾, 篠塚 圭祐, 二木 功治, 緒方 謙太郎, 橋口 明典

日本腎臓学会誌 （(一社)日本腎臓学会）  64 （ 6-E ） 563 - 563 2022年10月

共著,  ISSN  0385-2385

腎臓の間質にIgM-λ陽性の沈着物を認めた低悪性度B細胞リンパ腫の一例

川口 隆久, 唐澤 隆明, 橋元 奈津子, 橋口 明典, 熊谷 聡佑, 有馬 功一郎, 安藤 孝

神奈川腎炎研究会抄録集 （神奈川腎炎研究会）  79回   np93 - np114 2022年10月

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膜性腎症とMPO-ANCA関連血管炎を合併した高齢男性の1例

野間 悠太朗, 竜崎 正毅, 田島 敬也, 伊藤 亘, 松本 朋美, 坂口 知広, 森田 洋平, 貫井 紀宏, 池上 良, 橋口 明典, 久保田 英司

日本腎臓学会誌 （(一社)日本腎臓学会）  64 （ 6-E ） 570 - 570 2022年10月

共著,  ISSN  0385-2385

A girl with membranous nephropathy associated with ventriculoperitoneal shunt infection.

Awazu M, Miyahara M, Chiga M, Hashiguchi A, Takahashi H

CEN case reports （CEN case reports）  12 （ 1 ） 130 - 134 2022年09月

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Glomerulopathy associated with shunt infection is commonly membranoproliferative glomerulonephritis, whereas the causative organisms of secondary membranous nephropathy are usually viruses. We report a case of membranous nephropathy associated with shunt infection. The patient was born at 29-week gestation with a birth weight of 1178 g. Ventriculoperitoneal shunt surgery had been performed for congenital hydrocephalus. Thereafter, she had experienced seven shunt infections. At the age 13 years, proteinuria was detected in a school urinary screening. Urinalysis at our hospital demonstrated 3 + protein and 3 + blood. Laboratory testing demonstrated a serum creatinine 0.5 m/dl, albumin 2.5 g/dl, C-reactive protein (CRP) 13.7 mg/dl, and C3 182 mg/dl. Prior to repeat urinalysis, the patient developed vomiting and was admitted with suspected shunt infection. On admission, her body temperature was 36.0 ºC. Physical examination was unremarkable other than small stature and a palpable mass in the left upper quadrant. Urinalysis demonstrated 2 + protein and 1 + blood with no cells or casts. The urinary protein excretion was 3 g/day. Abnormal laboratory tests included erythrocyte sedimentation rate 102 mm/hr, CRP 11.67 mg/dl, IgG 2442 mg/dl, C3 177 mg/dl, and C4 44 mg/dl. Antibiotic therapy was initiated for a presumptive diagnosis of shunt infection and the shunt catheter was removed. Cultures obtained after antibiotic administration were negative. Proteinuria persisted after control of the shunt infection. Histology of a renal biopsy demonstrated membranous nephropathy with diffuse granular IgG staining and subepithelial deposits. Three possible pathomechanisms for her membranous nephropathy were considered.

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Structural modeling for Oxford histological classifications of immunoglobulin A nephropathy

Joh K., Nakazato T., Hashiguchi A., Shimizu A., Katafuchi R., Okonogi H., Koike K., Hirano K., Tsuboi N., Kawamura T., Yokoo T., Narita I., Suzuki Y.

PLoS ONE （PLoS ONE）  17 （ 9 September ） e0268731 2022年09月

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In immunoglobulin A nephropathy (IgAN), Cox regression analysis can select independent prognostic variables for renal functional decline (RFD). However, the correlation of the selected histological variables with clinical and/or treatment variables is unknown, thereby making histology-based treatment decisions unreliable. We prospectively followed 946 Japanese patients with IgAN for a median of 66 mo. and applied structural equation modeling (SEM) to identify direct and indirect effects of histological variables on RFD as a regression line of estimated glomerular filtration rate (eGFR) via clinical variables including amount of proteinuria, eGFR, mean arterial pressure (MAP) at biopsy, and treatment variables such as steroid therapy with/without tonsillectomy (ST) and renin-angiotensin system blocker (RASB). Multi-layered correlations between the variables and RFD were identified by multivariate linear regression analysis and the model's goodness of fit was confirmed. Only tubular atrophy/interstitial fibrosis (T) had an accelerative direct effect on RFD, while endocapillary hypercellularity and active crescent (C) had an attenuating indirect effect via ST. Segmental sclerosis (S) had an attenuating indirect effect via eGFR and mesangial hypercellularity (M) had accelerative indirect effect for RFD via proteinuria. Moreover, M and C had accelerative indirect effect via proteinuria, which can be controlled by ST. However, both T and S had additional indirect accelerative effects via eGFR or MAP at biopsy, which cannot be controlled by ST. SEM identified a systemic path links between histological variables and RFD via dependent clinical and/or treatment variables. These findings lead to clinically applicable novel methodologies that can contribute to predict treatment outcomes using the Oxford classifications.

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キャッスルマン病で治療中に壊死性筋膜炎及び急性腎障害を呈した2型糖尿病患者の1例

山口 慎太郎, 安達 京華, 吉本 憲史, 安田 格, 内山 清貴, 田島 敬也, 吉野 純, 橋口 明典, 神田 武志, 目黒 周, 伊藤 裕

糖尿病 （(一社)日本糖尿病学会）  65 （ 7 ） 395 - 395 2022年07月

共著,  ISSN  0021-437X

興味深い経過をたどった糖尿病性胃腸症の1例

鍛治場 寛, 村田 祥磨, 生形 晃男, 清水 彩, 北川 清宏, 野田 まりん, 久武 祐太, 野島 淳, 橋口 明典, 寺元 研, 東澤 俊彦, 関根 忠一, 岡村 幸重

日本消化器病学会関東支部例会プログラム・抄録集 （日本消化器病学会-関東支部）  370回   25 - 25 2022年07月

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Diagnosis of monoclonal immunotactoid glomerulopathy with positive λ chain by immunoelectron microscopy.

Sugita E, Sonoda H, Ryuzaki M, Hashiguchi A, Tokuyama H, Wakino S, Kanda T, Itoh H

CEN case reports （CEN case reports）  12 （ 1 ） 7 - 13 2022年06月

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We report the case of a 73-year-old-man who developed immunotactoid glomerulopathy (ITG). ITG is a rare disease characterized by proliferative glomerulonephritis and capillary wall deposits with a 10-60 nm diameter microtubular substructure. In monoclonal ITG, immunofluorescence analysis typically exhibits IgG with light chain restriction. Recent reviews recommend distinguishing monoclonal ITG from polyclonal ITG because monoclonal ITG is associated with a higher incidence of hematological disorders and better responsiveness to clone-directed therapy and renal prognosis. In our case, IgG, IgA, and IgM were negative by routine immunofluorescence; however, immunoelectron microscopy revealed positive λ chain. At 6 months after renal biopsy, the IgG λ chain was detected in the serum and urine, reflecting possible monoclonality. Therefore, it is useful to perform immunoelectron microscopy and follow-up with serum and urine protein electrophoresis and immunofixation to diagnose monoclonal ITG, even when routine immunofluorescence shows negative or nonspecific findings.

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MPO-ANCA、抗GBM抗体共陽性かつ膜性腎症を合併した急速進行性糸球体腎炎に対して選択的血漿交換(SePE)を行った一例

星 健太, 浦井 秀徳, 渡部 統, 中村 彰良, 橋口 明典, 村上 円人

日本急性血液浄化学会雑誌 （(NPO)日本急性血液浄化学会）  13 （ 1 ） 48 - 51 2022年06月

共著,  ISSN  2185-1085

　概要を見る

当院において、半月体形成性糸球体腎炎を呈したMPO-ANCA、抗糸球体基底膜(glomerular basement membrane:GBM)抗体共陽性かつ膜性腎症を合併した急速進行性糸球体腎炎(rapidly progressive glomerulonephritis:RPGN)を経験した。血清Crの上昇がみられた早期に副腎皮質ステロイド、静注シクロホスファミドパルス療法(intravenous cyclophosphamide:IVCY)に加え選択的血漿交換(selective plasma exchange:SePE)を行ったところ、2抗体の陰性化を得られ肺障害を生じることなく腎機能障害も改善した。発症早期に、免疫抑制療法に加えて凝固因子補充不要なSePEを行うことが新規抗体産生抑制および抗体除去に有用であると考えられた。(著者抄録)

【IgA腎症-最新の知見】腎生検組織標本へのAIの活用

橋口 明典

腎と透析 （(株)東京医学社）  92 （ 6 ） 1072 - 1076 2022年06月

共著,  ISSN  0385-2156

Clinical and pathological characteristics of patients with acute kidney injury in Japan in whom kidney biopsy was performed: a cross-sectional analysis of the Japan Renal Biopsy Registry (J‑RBR).

Moniwa N, Tanaka M, Sato H, Shimizu A, Sugiyama H, Yokoyama H, Tsuruya K, Hashiguchi A, Committee for Renal Biopsy and Disease Registry of the Japanese Society of Nephrology.

Clinical and experimental nephrology （Clinical and Experimental Nephrology）  26 （ 10 ） 963 - 973 2022年05月

共著,  ISSN  1342-1751

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Introduction: Acute kidney injury (AKI) is a worldwide concern and it leads to a poor prognosis or end-stage kidney disease. The purpose of this study was to clarify the characteristics of patients with AKI in whom kidney biopsy was performed using data of the Japan Renal Biopsy Registry (J-RBR). Methods: We screened 38,351 cases that were registered in the J-RBR from 2007 to 2018. We obtained data for 383 patients with AKI based on clinical diagnosis for analysis 1 and data for 714 patients with acute interstitial nephritis (AIN) or acute tubular necrosis (ATN) based on pathological diagnosis for analysis 2. Results: Of the cases screened, 383 patients with AKI (1.0%) were included in analysis 1. The main pathological diagnoses of AKI were AIN, ATN, chronic interstitial nephritis, nephro-sclerosis and crescentic glomerulonephritis. Of the cases screened, 589 patients with AIN (1.5%) and 110 patients with ATN (0.3%) were included in analysis 2. The main clinical diagnoses of AIN were AKI, rapidly progressive glomerulonephritis (RPGN), chronic nephritic syndrome (CNS) and drug-induced nephropathy (DIN), whereas those of ATN were AKI, RPGN, DIN and CNS. ATN patients had a higher serum creatinine level than that of AIN patients. Conclusion: Our results revealed that cases in the J-RBR included 1.0% of AKI cases based on clinical diagnosis and 1.5% and 0.3% of AIN and ATN cases, respectively, based on pathological diagnosis. In patients with suspected intrinsic AKI, kidney biopsy should be performed for diagnosis of the precise etiology and selection of appropriate treatment.

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無症候性血尿で経過観察中にSARS-CoV-2ワクチンによる肉眼的血尿を伴いIgA腎症と診断した2例

森澤 和美, 高橋 努, 濱田 陸, 幡谷 浩史, 本田 尭, 三上 直朗, 松岡 健太郎, 緒方 謙太郎, 山中 真理子, 尾原 健太郎, 橋口 明典, 本田 雅敬

日本小児腎臓病学会雑誌 （(一社)日本小児腎臓病学会）  35 （ 1Suppl. ） 156 - 156 2022年04月

共著,  ISSN  0915-2245

異なる撮像機のデジタルスライドに対して抗酸菌検出AIの再現性を得るための検討

江本 桂, 松田 紘典, 阿部 時也, 橋口 明典, 坂元 亨宇

日本病理学会会誌 （(一社)日本病理学会）  111 （ 1 ） 240 - 240 2022年03月

共著,  ISSN  0300-9181

当院で施行した移植腎生検病理組織の検討

坂巻 裕介, 香野 日高, 橋口 明典, 環 聡, 萩生田 純, 桝田 司, 宍戸 偉海, 井上 博之, 徳山 博文, 中川 健

日本臨床腎移植学会プログラム・抄録集 （(一社)日本臨床腎移植学会）  55回   288 - 288 2022年02月

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Quantitative evaluation of color difference between actual specimens and whole-slide imaging-scanned images calibrated with commercial color charts

Murayama Y., Sugiyama T., Ogino Y., Furuta H., Kambara T., Kamisono K., Abe T., Emoto K., Hashiguchi A., Kajimura Y., Sakamoto M.

Progress in Biomedical Optics and Imaging - Proceedings of SPIE （Progress in Biomedical Optics and Imaging - Proceedings of SPIE）  12039 2022年

共著,  ISSN  16057422

　概要を見る

When using different cameras and displays in the shot and display of one subject, different color images are often showed. To solve this problem, we have developed a color chart in which the constituent colors dyed with dyes are evenly distributed in the color space. We have also developed a tool that creates an International Color Consortium (ICC) profile from the captured image of this color chart. In this report, we will describe the color difference correction accuracy when our method is applied to actual stained pathological specimens taken with multiple whole-slide imaging (WSI). We confirmed color correction accuracy of Lab values in major parts such as the cell nucleus. The Lab value of the specimen itself measured by a spectrocolorimeter was compared with that of the captured image. As a result, the color difference ?E in H&E-stained cell nucleus was improved from 32.2 to 6.4 for Nanozoomer and from 13.5 to 7.2 for ultra-fast scanner (UFS) by our color correction. The results of the evaluations for other areas and other stain methods (PAS, EVG, MT, and PAM) were good. In the future, high-accuracy color correction of teacher data/evaluation data in AI diagnosis using pathological images will be important.

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