Laryngeal salivary duct carcinoma with pagetoid spread: a very rare case report


Aşkın R. R., Öztürk Sarı Ş., Şen C., Yegen G., Özlük M. Y.

37th European Congress of Pathology, Vienna, Avusturya, 6 Eylül - 10 Aralık 2025, cilt.487, sa.1, ss.400, (Özet Bildiri)

  • Yayın Türü: Bildiri / Özet Bildiri
  • Cilt numarası: 487
  • Basıldığı Şehir: Vienna
  • Basıldığı Ülke: Avusturya
  • Sayfa Sayıları: ss.400
  • İstanbul Üniversitesi Adresli: Evet

Özet

Background & Objectives: Paget disease is a rare carcinoma characterized by an intraepithelial growth of neoplastic cells originating in the skin or representing the intraepithelial spread of an underlying visceral carcinoma. It usually is located in the breast or apocrine-rich areas.

Methods: Here, we present a rare case of laryngeal salivary duct carcinoma (SDC) with pagetoid spread in a 71-year-old male. The patient had a 2-year history of hoarseness and new-onset stridor, which led to the decision to perform a tracheostomy, followed by a laryngeal biopsy. Further imaging revealed a 1.5 cm soft tissue lesion in the neck that infiltrates thyroid tissue, which was also biopsied, with no other masses identified elsewhere. Both biopsies were sent for histopathological examination with a one-week interval. Results: The first biopsy revealed intraepithelial carcinoma cells, with no additional lesions identified in the subepithelium, aside from some inflammatory cells. The second biopsy from the neck mass demonstrated neoplastic infiltration consisting of cells with eosinophilic cytoplasm and large, round, and pleomorphic nuclei, organized in irregular clusters within a desmoplastic stroma. Immunohistochemical analysis showed positivity for cytokeratin 7, EMA, CEA, and androgen receptor, while cytokeratin 20, p40, TTF-1, PAX8 and S100 were negative. Identical immunohistochemical findings were observed for both biopsies. Laryngoscopic findings showing submucosal mass localized in the subglottic area and histopathological findings supported a diagnosis of laryngeal SDC.

Conclusion: This case is particularly intriguing and rare for several reasons. Paget’s disease in the head and neck region is exceedingly uncommon, with the majority of reported cases involving the oral mucosa. Additionally, SDC, typically associated with major salivary glands, was found in the minor salivary glands of the larynx in this instance. To the best of our knowledge, this represents the first documented case of laryngeal SDC with pagetoid spread in the literature.