We report on the thyroid involvement with Langerhans cell histiocytosis (LCH) in a 3-year-old male. The patient presented with goiter and primary hypothyroidism. His goiter caused life-threatening airway obstruction. He developed locally invasive disease 4 years after his response to LCH therapy. LCH should be suspected as a cause of goiter and thyroidectomy is recommended.
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