Human papilloma virus (HPV) presence in primary tumors of the lacrimal sac: a case series and review of the literature

ABSTRACT

Primary tumors of the lacrimal sac (PTLS) are a rare subtype of ocular adnexa tumors, with potentially life-threatening clinical course. There has been growing evidence of human papilloma virus (HPV) as an etiological agent in these tumors.

In this retrospective observational case series, we report three cases of PTLS. All three underwent an initial dacryocystorhinostomy revealing a tissular mass in the lacrimal sac. Histological findings were respectively epithelial papilloma, epithelial Malpighian papilloma, and undifferentiated epidermoid carcinoma. PCR evaluation identified HPV serotype 6 in the first case and 16 in the third, and high p16 expression was found in the second case.

These three cases of PTLS with HPV detection complement 36 other cases identified in the literature, further incriminating HPV in the pathogenesis of these neoplasms. Ophthalmologists must remain wary of chronic lacrimal occlusion symptoms, and resort to CT scan and orbital Doppler sonography whenever first-line treatment fails.

KEYWORDS:

• Ocular adnexa neoplasm
• human papilloma virus
• lacrimal sac papilloma and carcinoma

Disclosure statement

The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the article.

Oscar Haigh has nothing to disclose.

Emmanuel Racy has nothing to disclose.

Corinne Bordonné has nothing to disclose.

Emmanuel Barreau has nothing to disclose.

Antoine Rousseau has been an occasional consultant on subjects outside the scope of this work for Thea, Horus Pharma, AbbVie, Alnylam, Glaukos, Santen, Bausch and Lomb.

Marc Labetoulle has been an occasional consultant on subjects outside the scope of this work for Alcon, AbbVie, Bausch and Lomb, Dompe, Horus, GSK, MSD, Novartis, Santen, Shire, and Thea.

Author contributions

All authors participated in the acquisition of data, designing the analyses, interpreting the results and writing the manuscript.

Data availability statement

The datasets generated during and/or analyzed during the current study are not publicly available, but are available from the current author on reasonable request.

Ethical statement

Ethical approval is not required for this study in accordance with local or national guidelines.