We were able to ascertain a history of chronic infections and osteomyelitis, hypoglycemia, and later a heterozygous genetic mutation in HYOU1 (exon 23, c.2638G>A) (p.Ala880Thr). We present the first case of a patient with an HYOU1 mutation associated with recurrent osteomyelitis and subglottic stenosis. This evidence concerns the gene HYOU1 and Hypoglycemia.