Discordant Phenotypes of Nephritis in Patients with X-linked Agammaglobulinemia

被引：0

作者：

Kanamori, Toru ^{[1
]}

Udagawa, Tomohiro ^{[1
]}

Fujii, Takayuki ^{[2
]}

Matsukura, Hiroyoshi ^{[3
]}

Iwaya, Yuka ^{[4
]}

Sonoda, Motoshi ^{[4
]}

Sugimoto, Keisuke ^{[5
]}

Takeguchi, Masahiro ^{[6
]}

Yoshino, Atsunori ^{[7
]}

Wang, I-Feng ^{[8
]}

Hwang, Daw-Yang ^{[8
]}

Schroeder, Harry W. ^{[9
]}

Shimizu, Masaki ^{[1
]}

Ochs, Hans D. ^{[10
]}

Morio, Tomohiro ^{[1
]}

Kanegane, Hirokazu ^{[11
]}

机构：

[1] Tokyo Med & Dent Univ TMDU, Grad Sch Med & Dent Sci, Dept Pediat & Dev Biol, 1-3-45 Yushima,Bunkyo Ku, Tokyo 1138519, Japan

[2] Seirei Sakura Citizen Hosp, Dept Nephrol, Sakura, Japan

[3] Saiseikai Takaoka Hosp, Dept Pediat, Takaoka, Japan

[4] Kyushu Univ, Grad Sch Med Sci, Dept Pediat, Fukuoka, Japan

[5] Kinki Univ, Fac Med, Dept Pediat, Osaka, Japan

[6] Oita Univ, Fac Med, Dept Pediat, Oita, Japan

[7] Dokkyo Med Univ, Saitama Med Ctr, Dept Pediat, Saitama, Japan

[8] Kaohsiung Med Univ, Natl Inst Canc Res, Natl Hlth Res Inst, Div Nephrol, Kaohsiung, Taiwan

[9] Univ Alabama Birmingham, Div Clin Immunol & Rheumatol, Birmingham, AL USA

[10] Univ Washington, Seattle Childrens Res Inst, Dept Pediat, Seattle, WA USA

[11] Tokyo Med & Dent Univ TMDU, Grad Sch Med & Dent Sci, Dept Child Hlth & Dev, 1-3-45 Yushima,Bunkyo Ku, Tokyo 1138519, Japan

来源：

JOURNAL OF CLINICAL IMMUNOLOGY | 2024年 / 44卷 / 07期

关键词：

X-linked agammaglobulinemia; Chronic glomerulonephritis; Tubulointerstitial nephritis; Leaky B cells; Immunoglobulin replacement therapy; IGA NEPHROPATHY; MANIFESTATIONS; CELLS;

D O I：

10.1007/s10875-024-01766-x

中图分类号：

R392 [医学免疫学]; Q939.91 [免疫学];

学科分类号：

100102 ;

摘要：

PurposeTo define the clinical and histological characteristics of nephritis in patients with X-linked agammaglobulinemia (XLA) and their immunological profiles.MethodsThe clinical, immunological, and histological findings of nine patients with XLA and nephritis were retrospectively analyzed.ResultsBased on kidney histological findings, patients with XLA and nephritis could be divided into two groups, viz., chronic glomerulonephritis (CGN) and tubulointerstitial nephritis (TIN). The two groups showed different immunological profiles. Patients in the CGN group exhibited an atypical immunological profile of XLA, with pathogenic leaky B cells producing immunoglobulins that may play a role in forming immune complexes and causing immune-mediated glomerulonephritis. In contrast, patients in the TIN group exhibited a typical immunological profile of XLA, suggesting that antibody-independent/other BTK-dependent mechanisms, or immunoglobulin replacement therapy (IgRT)-related immune/nonimmune-mediated nephrotoxicity causes TIN.ConclusionNephritis occurring in patients with XLA could have links between their renal pathology and immunological status. Careful observation is recommended to detect kidney pathology in patients with XLA on IgRT.

引用

页数：8

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