著者

Satoshi Kurahashi Naohiro Toda Masaaki Fujita Katsuya Tanigaki Jun Takeoka Hisako Hirashima Eri Muso Katsuhiro Io Takaki Sakurai Toshiyuki Komiya

出版者

The Japanese Society of Internal Medicine

雑誌

Internal Medicine (ISSN:09182918)

巻号頁・発行日

pp.8046-21, (Released:2021-09-18)

参考文献数

20

被引用文献数

1

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Rosai-Dorfman-Destombes disease (RDD) is a non-Langerhans cell histiocytosis characterized by the accumulation of histiocytes inside the lymph nodes or extranodally. The association between RDD and IgG4-related disease (IgG4-RD) is discussed. We herein report a case of RDD manifesting as acute tubulointerstitial nephritis mimicking IgG4-RD. The first renal biopsy showed severe tubulointerstitial nephritis with infiltration of S100-positive histiocytes and IgG4-positive plasma cells; storiform fibrosis and obliterative phlebitis were not confirmed. After prednisolone therapy, IgG4-positive cells and S100-positive histiocytes were decreased, but the IgG4/IgG ratio increased despite clinical improvement. These findings indicated extranodal RDD in the kidney presenting as tubulointerstitial nephritis.

著者

Shanika Nanayakkara STMLD Senevirathna Tilak Abeysekera Rohana Chandrajith Neelakanthi Ratnatunga EDL Gunarathne Junxia Yan Toshiaki Hitomi Eri Muso Toshiyuki Komiya Kouji H. Harada Wanyang Liu Hatasu Kobayashi Hiroko Okuda Hideyuki Sawatari Fumihiko Matsuda Ryo Yamada Takao Watanabe Hideki Miyataka Seiichiro Himeno Akio Koizumi

出版者

Japan Society for Occupational Health

雑誌

Journal of Occupational Health (ISSN:13419145)

巻号頁・発行日

pp.13-0172-OA, (Released:2013-12-18)

被引用文献数

91

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Objectives: Previous investigations on chronic kidney disease of unknown etiology characterized by tubulointerstitial damages (CKDu) in the North Central Region (NCR) of Sri Lanka have supported the involvement of social, environmental and genetic factors in its pathogenesis. Methods: We conducted a social-environmental-and-genetic epidemiology study on a male population in NCR to investigate the genetic and environmental contributors. We recruited 311 case-series patients and 504 control candidates. Of the 504 control candidates, 218 (43%) were eliminated because of the presence of hypertension, proteinuria, high HBA1c, high serum creatinine or high alpha-1 microglobulin in urine. Results and Discussion: None of 18 metals measured (µg/l) in urine, including Cd, As and Pb, showed significantly higher concentrations in cases compared with controls. As speciation results showed that 75-80% of total urinary As was in the form of arsenobetaine, which is non-toxic to humans. None of the metal concentrations in drinking water samples exceeded guideline values. A genome-wide association study (GWAS) was conducted to determine the genetic contributors. The GWAS yielded a genome-wide significant association with CKDu for a single nucleotide polymorphism (SNP; rs6066043; P=5.23×10-9 in quantitative trait locus analysis; P=3.73×10-8 in dichotomous analysis) in SLC13A3 (sodium-dependent dicarboxylate transporter member 3). The population attributable fraction and odds ratio for this SNP were 50% and 2.13. Genetic susceptibility was identified as the major risk factor for CKDu. However, 43% of the apparently healthy male population suffers from non-communicable diseases, suggesting their possible influence on CKDu progression.