Regional Spread of Recurrent Respiratory Papillomatosis to Bilateral Cervical Lymph Nodes
Recurrent respiratory papillomatosis (RPR) is a chronic disease that affects both pediatric and adult populations. It is caused by human papillomavirus (HPV) types 6 and 11 and is characterized by a benign proliferation of squamous epithelium along the upper aerodigestive tract. The disease is classified into juvenile versus adult onset and has a clinical course that may be benign or aggressive. Benign disease is usually limited to the larynx and is characterized by few or no recurrences.
... recurrences. Aggressive disease is characterized by epithelial atypia and multiple recurrences with disease spread to other sites within the upper aerodigestive tract. The glottic larynx is considered the most common site affected, however, RPR may involve any site along the aerodigestive tract including the oral cavity and soft palate. Although there is a risk of malignant transformation in RPR, it is considered a benign disease. Similarly, papillomas found within the nasal cavity and, by extension, the paranasal sinuses are considered viral-related papillomas where the benign squamous proliferation involves the Schneiderian epithelium of the nasal cavity. As with RPR, Schneiderian papillomas are benign tumors with a predilection for local recurrence. Schneiderian papillomas are divided into fungiform, inverted and cylindrical types based on the specific architectural patterns seen histologically. As with RPR, Schneiderian papilloma is a benign condition and is not known to metastasize in the absence of malignant transformation. We present a case of benign recurrent papillomatosis involving the upper aerodigestive tract with regional metastases to bilateral cervical lymph nodes. A review of the literature reveals this case to be only the third reported case of benign papillomatous metastasis to the neck. A 47-year-old man presented to our clinic with a longstanding history of adultonset recurrent respiratory papillomatosis involving his nasal cavity, nasopharynx, oral cavity, oropharynx and supraglottic larynx ( Figure 1 ). He had previously undergone multiple excisions of his lesions with pathology consistent with Schneiderian papilloma-type lesions with no evidence of malignancy. He presented with a new 3 x 2 cm right level II neck mass which had been present over the last few months. The mass was asymptomatic but had slightly increased in size over the previous few months. The patient was taken to the operating room where he underwent direct laryngoscopy with excision of the right neck mass. Intraoperatively, he was noted to have typical-appearing papillomas along his palate and posterior oropharyngeal wall without extension into the larynx. The right neck mass was noted to be well-encapsulated with a soft, yellow cheese-like material within it. Pathology results of the right neck mass were consistent with a lymph node containing cystadenomatous foci and epithelium resembling exophytic or fungiform Schneiderian papilloma. There was no evidence of carcinoma. Polymerase chain reaction studies of the lymph node revealed the presence of HPV type 11. Of note, four years prior, the patient had undergone a lymph node excision on the contralateral neck with pathology consistent with benign papillomatosis.