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HPV-related sinonasal carcinoma with adenoid cystic-like features with intracranial invasion.

A 60-year-old man was referred to the Department of Otolaryngology for evaluation of a right nasal tumor. He had a 3-month history of intermittent right epistaxis. One week before admission, he had noticed a sudden loss of vision in his right eye while watching television. At that time, he attended the outpatient clinic of the Department of Ophthalmology. Ophthalmologic examination showed limited bilateral eye motion in all directions, right ptosis, and a loss of vision in the right eye. No other neurologic signs were observed. Magnetic resonance imaging demonstrated a sinonasal tumor with orbital and intracranial extension (figure 1). He was referred to the Department of Otolaryngology for further management.

Anterior rhinoscopy showed a huge mass over the right nasal cavity, and nasopharyngoscopy revealed a smooth nasopharynx surface. All other otolaryngologic examinations were normal. A nasal biopsy was performed, revealing a sinonasal teratocarcinosarcoma. After discussions with the patient, debulking surgery combined with radiotherapy was suggested and performed on the second day after admission.

Histology of the sinonasal tumor retrieved after endoscopic debulking surgery revealed infiltration of solid sheets of basaloid cells with vesicular nuclei, nucleoli, and mitoses separated by fibrous bands of collagenized stroma. Focal microcystic spaces with basophilic material resembling adenoid cystic carcinoma were noted (figure 2, A). Immunohistochemistry staining was positive for p16 (a marker for human papillomavirus [HPV] infection) (figure 2, B); the genotyping was HPV-14D.

Based on these findings, a diagnosis of HPV-related sinonasal carcinoma with adenoid cystic-like features was provided by the pathologist. No obvious complications, such as bleeding, meningitis, or sepsis, were noted postsurgically. Nevertheless, the patient did not recover the vision in his right eye.

The patient was discharged on the ninth day after admission. A radiotherapy plan of 7,000 cGy/35 fx was followed. To date, the patient continues follow-up at the outpatient clinic. Radiotherapy decreased the size of the tumor, but the patient's vision in his right eye did not return. Intermittent epistaxis was noted during follow-up, and the patient died 11 months postsurgically.

Sinonasal tumors are rare, with an annual incidence of approximately 1 case per 100,000 inhabitants worldwide. (1) However, with the development of endoscopic sinus surgery and improvements in histologic detection, new sinonasal lesions are being diagnosed more often. When Bishop et al researched the incidence and clinicopathologic profile of HPV-related carcinomas of the sinonasal tract, a variant resembling adenoid cystic carcinoma was found. (2,3) HPV-related sinonasal carcinoma with adenoid cystic-like features is a recently described tumor with less than 10 cases reported in the literature to date. (2-4)

In rare instances, a sudden onset of visual loss has been reported in patients with pituitary adenoma, craniopharyngioma, mucoceles, and pyoceles of the paranasal sinuses, as well as in sphenoid sinus esthesioneuroblastoma. (5) The case reported herein emphasizes that HPV-related sinonasal carcinoma with adenoid cystic-like features is one cause of sinonasal tumors, and that it may lead to sudden blindness and intermittent epistaxis.

From the Department of Otorhinolaryngology-Head and Neck Surgery (Dr. Hung, Dr. Chung, and Dr. Guo) and the Department of Pathology (Dr. Chen), Changhua Christian Hospital, Changhua City, Taiwan.

References

(1.) Llorente JL, Lopez F, Suarez C, Hermsen MA. Sinonasal carcinoma: Clinical, pathological, genetic and therapeutic advances. Nat Rev Clin Oncol 2014;! l(8):460-72.

(2.) Bishop JA, Ogawa T, Stelow EB, et al. Human papillomavirus-related carcinoma with adenoid cystic-like features: A peculiar variant of head and neck cancer restricted to the sinonasal tract. Am J Surg Pathol 2013;37(6):836-44.

(3.) Bishop JA, Guo TW, Smith DF, et al. Human papillomavirus-related carcinomas of the sinonasal tract. Am J Surg Pathol 2013;37(2):185-92.

(4.) Wenig BM. Recently described sinonasal tract lesions/neoplasms: Considerations for the new World Health Organization book. Head Neck Pathol 2014;8(1):33-41.

(5.) Tamhankar MA, Volpe NJ, Loevner LA, et al. Primary sinonasal undifferentiated carcinoma presenting with bilateral retrobulbar optic neuropathy. J Neuroophthalmol 2007;27(3):189-92.

Caption: Figure 1. Coronal MR] of the nasal cavity shows a sinonasal tumor with orbital and intracranial extension.

Caption: Figure 2. A: Focal microcystic spaces with basophilic material resembling adenoid cystic carcinoma are noted. B: The specimen is positive for pl6 immunohistochemistry staining.
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Title Annotation:PATHOLOGY CLINIC
Author:Hung, Yuntsung; Chung, Yu-Yen; Chen, Mei-Ling; Guo, Jyun-Ying
Publication:Ear, Nose and Throat Journal
Geographic Code:1USA
Date:Jul 1, 2018
Words:694
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