Background: The clinical presentation of Langerhans cell histiocytosis (LCH) as a primary solitary nodule in the thyroid gland is rare. As a result, there are few reports of its cytologic features in thyroid aspirates where it can pose a diagnostic pitfall. Case and Conclusion: To foster familiarity with its cytomorphology, we report the fine-needle aspiration biopsy (FNAB) findings of 3 specimens from 2 patients with LCH presenting as a solitary thyroid nodule. All aspirates contained numerous dispersed cells with prominent nuclear grooves, and the background showed a mixed pattern of chronic inflammation including scattered eosinophils. The aspirate from patient 1 raised a differential diagnosis that included chronic lymphocytic thyroiditis and a thyroglossal duct cyst, while the aspirate from patient 2 was interpreted as ‘suspicious for papillary thyroid carcinoma'. The diagnosis of LCH was confirmed in both patients after lobectomy and immunohistochemical studies that revealed positive reactivity for CD1a and S-100. LCH of the thyroid gland is rare and can pose significant diagnostic challenges, but increased familiarity with its characteristic cytomorphology can help in avoiding diagnostic pitfalls.

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