J Clin Immunol (2017) 37:514–516 DOI 10.1007/s10875-017-0414-6 LETTER TO EDITOR 1 2 1 3 Amir B. Orandi & Tiphanie P. Vogel & Molly P. Keppel & Elizabeth C. Utterson & 1,4 Megan A. Cooper Received: 3 April 2017 /Accepted: 15 June 2017 /Published online: 22 June 2017 Springer Science+Business Media, LLC 2017 To the editor, respectively. She was subsequently treated with corticoste- We report the case of a 5-year-old Caucasian girl with very roids and azathioprine, with testing demonstrating wildtype early-onset inflammatory bowel disease (VOIBD) who had thiopurine methyltransferase genotype, predictive of normal inadequate response to initial therapy with azathioprine. azathioprine metabolism. Over the ensuing 12 months, she continued to have active disease and azathioprine was incre- Immune system evaluation revealed an isolated complete nat- ural killer cell (NK cell) deficiency, which resolved with dis- mentally increased to 3 mg/kg. Drug levels at this dose continuation of the drug. showed sub-therapeutic 6-TGN at 229 pmol/RBC (range The patient first presented at age 4 with a history of angular 230–400), below risk levels for myelotoxicity with no eleva- cheilitis and oral ulcerations of the soft palate. She had notable tion of the hepatotoxic metabolite 6-MMPN. findings of microcytic anemia (Hb
Journal of Clinical Immunology – Springer Journals
Published: Jun 22, 2017
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