Evidence map›Paper›PMID 39190146›Full record

ArticlePediatric nephrology (Berlin, Germany)2025

A rare case of late-onset immune dysregulation, polyendocrinopathy, enteropathy, X-linked (IPEX) syndrome confused with IgA vasculitis nephropathy.

Ruochen Che, Mengqiu Miao, Guixia Ding, Sanlong Zhao

Abstract readCase Reports
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In one paragraph

Article in Pediatric nephrology (Berlin, Germany), 2025. 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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4 · The record

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5 · Who and what money

Authors and funding

4 authors.

Ruochen CheDepartment of Nephrology, Children's Hospital of Nanjing Medical University, 72 Guangzhou Road, Nanjing, 210029, Jiangsu Province, China.
Mengqiu MiaoDepartment of Nephrology, Children's Hospital of Nanjing Medical University, 72 Guangzhou Road, Nanjing, 210029, Jiangsu Province, China.
Guixia DingDepartment of Nephrology, Children's Hospital of Nanjing Medical University, 72 Guangzhou Road, Nanjing, 210029, Jiangsu Province, China.
Sanlong ZhaoDepartment of Nephrology, Children's Hospital of Nanjing Medical University, 72 Guangzhou Road, Nanjing, 210029, Jiangsu Province, China. slzhao1981@163.com.

Funding

Health Science and Technology Development Special Foundation of Nanjing YKK22159National Natural Science Foundation of China 82200744
6 · The paper itself

Abstract

A 3-year-old boy initially presented with purpura-like rashes and nephrotic syndrome, suspected to be IgA vasculitis nephritis (IgAVN). The suggestion of kidney biopsy was rejected. Although the patient responded well to glucocorticoids, they later developed recurrent proteinuria, refractory diarrhea, and subsequent metabolic acidosis. Kidney biopsy showed membranous nephropathy with positive semaphorin 3B expression, indicative of other kidney diseases rather than IgAVN. Although his kidney responded well to glucocorticoid combined with cyclosporine A treatment regimen, enteropathy and severe food allergy still progressed afterwards as evidenced by villous atrophy on gastrointestinal endoscopy examination. Whole exome sequencing identified a heterozygous missense variant in exon 11 of FOXP3: c.1121 T > G, confirming the diagnosis of immune dysregulation, polyendocrinopathy, enteropathy, X-linked (IPEX) syndrome. The case expanded the phenotypic spectrum of IPEX syndrome, suggesting high phenotypic heterogeneity despite similar genotypes. It also put emphasis on the significance of kidney biopsy to differentiate IgA vasculitis nephropathy from other immune disorders.

Indexed as

Forkhead Transcription FactorsGenetic Diseases, X-LinkedIgA VasculitisImmune System DiseasesBiopsyChild, PreschoolDiabetes Mellitus, Type 1Diagnosis, DifferentialDiarrheaEndocrine System DiseasesExome SequencingFailure to ThriveGlomerulonephritis, IGAGlucocorticoidsHumansIntestinal DiseasesForkhead Transcription FactorsFOXP3 protein, humanGlucocorticoidsFOXP3IgA vasculitisIPEX syndromeMembranous nephropathy

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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.