Comments on “Langerhans cell histiocytosis of the lung and thyroid, co-existing with papillary thyroid cancer” Jamaati HR, Shadmehr MB, Saidi B, Khosravi A, Arab M, Mohammadi F. Endocr Pathol 20:133–136, 2009 and “Incidental langerhans cell histiocytosis of thyroid: case report and review of the literature” Foulet-Roge A, Josselin N, Guyetant S, Gardet JJ, Besancon A, Saint-Andre JP, Fabiani B. Endocr Pathol 13:227–233, 2002. We read with great interest the contributions of Jamaati et al. and Foulet-Roge et al. respectively entitled “Langerhans cell histiocytosis of the lung and thyroid, co-existing with papillary thyroid cancer” and “Incidental langerhans cell histiocytosis of thyroid: case report and review of the literature” [1, 2]. We have had a similar case of coexistent thyroid Langerhans cell histiocytosis (LCH) and papillary thyroid carcinoma (PTC). A 29-year-old male patient presented with a huge neck mass, immobile, and compressive, involving the trachea, the pharynx, the left jugular vein and the left carotid sheath. Bone (mandible), lung, skin and hypothalamo-pituitary LCH lesions had been documented for 6 years, and clinical stability had been obtained with corticosteroids. The transpharyngeal biopsies performed during a panendoscopy revealed a PTC with positive immunostaining for TTF1, HBME1 and CK19. Associated LCH involvement was diagnosed by the presence of Langerhans cells reactive to CD1a and S100 protein (Fig. 1). The cytological examination of a blind transcutaneous thyroid fine needle aspiration biopsy and other surgical biopsies isolated only the LCH section and a lymphocytic thyroiditis. A surgical resection of this advanced disease could not be considered and a multidisciplinary panel proposed a medical treatment (Prednisone 40 mg/day). Given that there was no response 6 months later, chemotherapy with Vinblastine (Velbe® 6 mg/m every 2 weeks) produced a stabilization over 14 months. Then, due to a severe increase in size and the extension from the oropharynx to the mediastinum in a vertical axis and from the cervical rachis to the subcutaneous tissues in a horizontal axis, a treatment with Cladribine was initiated (Leustatin® 0.1 mg/kg/day over 5 days every 2 months). After 7 months without a response, the treatment was stopped and changed to Imatinib (Gleevec® 100 mg/day for 1 month and then 400 mg/day). Three months after the beginning of this targeted therapy and 3 years after the appearance of the cervical mass, a dramatic dyspnea caused by the compression and the infiltration of the trachea occurred. A tracheostomy under local anaesthesia was carried out in difficult technical conditions. Despite this attempt, acute pulmonary distress resulted in the death of the patient. The family refused an autopsy. S. Vergez (*) Department of Head and Neck Surgery, University-Hospital Rangueil-Larrey, 24 chemin de Pouvourville, 31059 Toulouse Cedex 9, France e-mail: vergez.s@chu-toulouse.fr
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