Evidence map›Paper›PMID 42369267›Full record

ArticleKidney medicine2026

Early Tonsillectomy for Dual Remission of Periodic Fever, Aphthous Stomatitis, Pharyngitis, and Adenitis Syndrome and Immunoglobulin A Nephropathy: A Case Report.

Daishi Hirano, Kensuke Joh, Shiko Honma, Ryoko Sakaguchi, Aya Saito, Yuna Hiwatari, Shiho Yoshizawa, Yosuke Shoji, Haruhide Sakaguchi, Chisato Umeda and 2 more

Abstract readCase Reports
In one paragraph

Article in Kidney medicine, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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1 · What the graph read from it

What it found

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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

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3 · Its place in the literature

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0 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

12 authors.

Daishi HiranoDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Kensuke JohDepartment of Pathology, The Jikei University School of Medicine, Tokyo, Japan.
Shiko HonmaDepartment of Pathology, The Jikei University School of Medicine, Tokyo, Japan.
Ryoko SakaguchiDepartment of Pathology, The Jikei University School of Medicine, Tokyo, Japan.
Aya SaitoDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Yuna HiwatariDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Shiho YoshizawaDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Yosuke ShojiDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Haruhide SakaguchiDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Chisato UmedaDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Saori MiwaDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.
Akira ItoDepartment of Pediatrics, The Jikei University School of Medicine, Tokyo, Japan.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Despite sharing tonsillar immune dysregulation as a common pathogenic mechanism, concurrent presentation of periodic fever, aphthous stomatitis, pharyngitis, and adenitis (PFAPA) syndrome with immunoglobulin A (IgA) nephropathy (IgAN) remains exceptionally rare, with only 1 previous case reported in the literature receiving multidrug immunosuppressive therapy before tonsillectomy. In this study, we report the first pediatric case in which both conditions achieved sustained remission following early tonsillectomy without additional immunosuppression. A 6-year-old boy with a 3-year history of PFAPA developed gross hematuria coinciding with febrile episodes. Kidney biopsy confirmed IgAN. Owing to the poor response to cimetidine and significant disruption to schooling, palatine tonsillectomy was performed at 7 years. Following surgery, PFAPA episodes completely resolved, and hematuria ceased. Repeat kidney biopsy at 11 months postoperatively showed marked histological improvement, with resolution of mesangial hypercellularity and reduced endocapillary proliferation. This case provides the first pediatric case demonstrating that early tonsillectomy alone can effectively treat coexisting PFAPA and IgAN without the need for additional immunosuppressive therapy. The simultaneous clinical and histological remission supports a shared pathogenic mechanism involving tonsillar immune dysfunction and highlights tonsillectomy as a potentially curative intervention for select patients with concurrent tonsil-driven diseases.

Indexed as

focal infectionIgA nephropathypediatric autoinflammatory diseasePFAPA syndromesimultaneous remissionTonsillectomy

Identifiers

PMID42369267
PMCPMC13293688

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.