Atypical X‐Linked Severe Combined Immunodeficiency in a Patient With Early‐Onset Inflammatory Bowel Disease

Primary immunodeficiency disorders may present with gastrointestinal inflammation resembling inflammatory bowel disease (IBD), particularly in patients with pediatric or very-early-onset disease. We report a 24-year-old male with lifelong immune dysregulation and early-onset IBD who was ultimately found to carry a hemizygous intronic IL2RG variant (c.270-14C>G). Despite extensive infectious complications, lymphoproliferative disease, and chronic lung pathology, his Crohn's-like intestinal inflammation had been managed successfully with azathioprine. Genome sequencing followed by RNA analysis demonstrated that the IL2RG variant introduced a cryptic splice acceptor site, causing aberrant splicing, intron retention, and a premature stop codon, confirming a diagnosis of atypical or hypomorphic X-linked severe combined immunodeficiency (X-SCID). This case expands the phenotypic spectrum of X-SCID to include chronic IBD-like enterocolitis and underscores the diagnostic utility of comprehensive genomic and transcriptomic testing in patients with complex, unexplained immune and gastrointestinal disease. Recognition of atypical presentations of X-SCID has implications for surveillance, prognosis, and consideration of definitive therapies.

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Publication Details

Journal
American Journal of Medical Genetics Part A
Published
2026-11-02
DOI
https://doi.org/10.1002/ajmg.a.70352
Primary Topic
Immunodeficiency and Autoimmune Disorders
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article
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article

Atypical X‐Linked Severe Combined Immunodeficiency in a Patient With Early‐Onset Inflammatory Bowel Disease

Jennah C. Foltz, Jean Pappas Molleston, Theodore E. Wilson, Morgan N. Driver et al.
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American Journal of Medical Genetics Part A
Immunodeficiency and Autoimmune Disorders
article

Atypical X‐Linked Severe Combined Immunodeficiency in a Patient With Early‐Onset Inflammatory Bowel Disease

Jennah C. Foltz, Jean Pappas Molleston, Theodore E. Wilson, Morgan N. Driver, Agustina Fainguersch, Casey A. Thornton, Xiaoyi Zhang, Xiaonan Zhao, Sydney Lau, Jay Jun‐Hong Jin
article en

Abstract

Primary immunodeficiency disorders may present with gastrointestinal inflammation resembling inflammatory bowel disease (IBD), particularly in patients with pediatric or very-early-onset disease. We report a 24-year-old male with lifelong immune dysregulation and early-onset IBD who was ultimately found to carry a hemizygous intronic IL2RG variant (c.270-14C>G). Despite extensive infectious complications, lymphoproliferative disease, and chronic lung pathology, his Crohn's-like intestinal inflammation had been managed successfully with azathioprine. Genome sequencing followed by RNA analysis demonstrated that the IL2RG variant introduced a cryptic splice acceptor site, causing aberrant splicing, intron retention, and a premature stop codon, confirming a diagnosis of atypical or hypomorphic X-linked severe combined immunodeficiency (X-SCID). This case expands the phenotypic spectrum of X-SCID to include chronic IBD-like enterocolitis and underscores the diagnostic utility of comprehensive genomic and transcriptomic testing in patients with complex, unexplained immune and gastrointestinal disease. Recognition of atypical presentations of X-SCID has implications for surveillance, prognosis, and consideration of definitive therapies.

American Journal of Medical Genetics Part A
Baylor College of Medicine (US), Baylor Genetics (US), Indiana University Indianapolis (US), Indiana University School of Medicine (US)
Good health and well-being
Openalex Percentile: Top 20%
Immunodeficiency and Autoimmune Disorders
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Atypical X‐Linked Severe Combined Immunodeficiency in a Patient With Early‐Onset Inflammatory Bowel Disease — Jennah C. Foltz, Jean Pappas Molleston, et al. · American Journal of Medical Genetics Part A (2026) | TGRS Research Map | TGRS