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Cribriform Adenocarcinoma of Minor Salivary Gland Origin Principally Affecting the Tongue: Characterization of New Entity

Skalova, Alena MD, PhD*; Sima, Radek PhD; Kaspirkova-Nemcova, Jana Mgr; Simpson, Roderick H.W. MD; Elmberger, Goran MD§; Leivo, Ilmo MD, PhD; Di Palma, Silvana MD; Jirasek, Tomas MD, PhD; Gnepp, Douglas R. MD**; Weinreb, Ilan MD††; Perez-Ordoñez, Bayardo MD††; Mukensnabl, Petr MD, PhD*; Rychly, Boris MD‡‡; Hrabal, Petr MD§§; Michal, Michal MD*

The American Journal of Surgical Pathology: August 2011 - Volume 35 - Issue 8 - p 1168–1176
doi: 10.1097/PAS.0b013e31821e1f54
Original Articles

We present a series of 23 cases of a distinctive, hitherto poorly recognized low-grade adenocarcinoma, with several histologic features reminiscent of papillary carcinoma of the thyroid, and which mostly but not exclusively occurs in the tongue. All the tumors were unencapsulated and were divided into lobules that were composed mainly of cribriform and solid growth patterns. Therefore, we propose the name “cribriform adenocarcinoma of minor salivary gland origin (CAMSG).” All the patients were adults with a mean age at diagnosis of 55.8 years (range, 25 to 85 y). Fourteen of the 23 tumors were localized in the tongue, 3 in the soft palate, 2 in the retromolar buccal mucosa, 3 in the lingual tonsils, and 1 in the upper lip. Fifteen patients of 23 had synchronous metastases in the cervical lymph nodes at the time of diagnosis, bilateral in 3 cases. In 3 patients, the nodal metastasis was the first evidence of disease, later investigation revealing primary neoplasms in the base of tongue and tonsil, respectively. In addition, 1 patient developed a cervical lymph node metastasis 8 years after excision of a primary tumor of the tongue. Data on treatment and follow-up were available in 14 cases. The patients were treated by radical excision with clear margins (12 cases) or by simple excision (2 cases). Neck dissection was performed in 10 patients; 9 received radiotherapy, but none were treated by chemotherapy. Clinical follow-up ranged from 2 months to 13 years (mean, 6 y and 5 mo). Twelve patients are alive with no evidence of recurrent or metastatic disease after treatment, 1 patient died 2 years after surgery without evidence of tumor, and 1 patient is alive with recurrent tumor of the palate.

*Department of Pathology, Charles University Prague, Faculty of Medicine in Pilsen

Molecular Pathology Laboratory, Department of Pathology, Medical Faculty Hospital, Pilsen

Department of Pathology, Faculty Hospital Kralovske Vinohrady

§§Department of Pathology, Central Military Hospital, Prague, Czech Republic

Department of Histopathology, Royal Devon and Exeter Hospital, Exeter

Department of Histopathology, the RSCH, Division of Clinical Medicine University of Surrey, Guildford, Surrey, England

§Department of Pathology and Cytology, Karolinska University Hospital, Solna and Danderyd, Stockholm, Sweden

Department of Pathology, University of Turku, Turku, Finland

**Department of Pathology, Warren Alpert School of Medicine at Brown University, Rhode Island Hospital, Providence, RI

††Department of Pathology, University Health Network, University of Toronto, Toronto, Ontario, Canada

‡‡Cytopathos, Bratislava, Slovakia

Supported by: Grant Nr. 9725 of IGA MH CR (Internal Grant Agency of Health Ministry, Czech Republic).

The preliminary results of the study were presented at the SYMPOSIUM Updates in Head and Neck Pathology: at the XXVIIIth Congress of the International Academy of Pathology - São Paulo, Brazil, Oct 10th-15th 2010 (AS) and as a platform presentation at USCAP Meeting 2011, San Antonio, Feb 26th-March 4th 2011 (AS).

Correspondence: Alena Skálová, MD, PhD, Sikl's Department of Pathology, Medical Faculty of Charles University, Faculty Hospital, E. Benese 13, 305 99 Pilsen, Czech Republic (e-mail: skalova@fnplzen.cz).

© 2011 Lippincott Williams & Wilkins, Inc.