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.
Authors
- Jennah C. Foltz (ORCID: https://orcid.org/0009-0008-6381-5430)
- Jean Pappas Molleston (ORCID: https://orcid.org/0000-0002-8708-0298)
- Theodore E. Wilson
- Morgan N. Driver (ORCID: https://orcid.org/0000-0001-6738-0667)
- Agustina Fainguersch
- Casey A. Thornton (ORCID: https://orcid.org/0000-0003-3589-6911)
- Xiaoyi Zhang (ORCID: https://orcid.org/0000-0002-4988-819X)
- Xiaonan Zhao
- Sydney Lau
- Jay Jun‐Hong Jin
Institutions
- Baylor College of Medicine (US)
- Baylor Genetics (US)
- Indiana University Indianapolis (US)
- Indiana University School of Medicine (US)
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
- Type
- article
- Field-Weighted Citation Impact
- 0.00