Case report

A 45-year-old male patient reported with a chief complaint of an intra-oral growth on the left side of the palate for the past 6 months which was increasing in size ( Fig. 1 ). He gave a history of surgery carried out to his left nose 2 years previously with a biopsy report showing inverted type SPs. Intra-oral examination revealed an ulcerating proliferating mass extending from 25 to the maxillary tuberosity region with expansion of the palate, not crossing the midline. Growth was also noticed in relation to the left side of the nose. No lymph nodes were palpable. Computed tomography showed a homogenously enhancing mass involving the entire left maxilla, nasal cavity, extending to the ethmoids ( Fig. 2 ). Following the previous biopsy report, the patient was admitted for surgery, as the growth was considered to be a recurrent lesion. Sub-total maxillectomy with preservation of the orbital floor along with lateral rhinotomy was performed through a Weber–Fergusson incision. The histopathological diagnosis was SP, inverted type, with malignant transformation ( Fig. 3 ). 2 weeks after surgery the patient developed neck nodes in the level II region. Functional neck dissection clearing nodes from level I to V was performed. Histopathology revealed level II B and III were positive for tumour infiltration. Post-surgically the patient underwent radiotherapy. The patient is being followed up and has no evidence of disease.

Intra oral lesion.

CT scan showing the lesion involving the left maxilla.

Higher magnification showing papillary projections growing into the connective tissue. With areas showing proliferating strands and cords of squamous epithelium with nuclear atypia and dysplastic features such as hyperchromatic nuclei, individual cell keratinization and keratin pearl formation (haematoxylin–eosin stain, original magnification, ×40).

Discussion

In 1854, W ard first described schneiderian papillomas of the nose. These lesions were named after C. Victor Schneider who, in the 1600s, identified its origin from the ectoderm . R ingertz was the first to identify the tendency of SPs to invert into the underlying connective tissue stroma, which differs from other types of papillomas . Viruses have long been suspected to cause these neoplastic lesions because they have a well-known tendency to produce papillomas elsewhere in the body. Human papilloma virus (HPV) is an epitheliotropic virus that has been implicated in premalignant and malignant lesions of the anogenital tract. Similarly, both the low-risk subtypes (HPV 11, HPV 6) and the high-risk subtypes (HPV 16, HPV 18) have been identified in SPs . SCC is the most common malignant neoplasm associated with SPs. Other types of malignancy rarely associated with SPs are adenocarcinoma and small cell carcinoma . Of the 3 subtypes of SPs, fungiform papillomas have not been reported to have malignant potential . IPs have been reported to develop into carcinoma in 5–10% of cases . Cylindrical papillomas appear to have a higher frequency (14–19%) of malignant association. Approximately 10% of IPs harbour SCC . The recurrence rate of these neoplastic lesions is highly variable (0–78%), depending mainly on the type of surgical approach and the completeness of resection .

The recurrence rate after lateral rhinotomy and maxillectomy is low compared with transnasal excision with the Caldwell–Luc operation (35%) or non-endoscopic transnasal excision alone (58%) . The multicentric origin of SPs has also been proposed as another factor that leads to the high recurrence rate, but this has been documented in only a few cases . Unilateral nasal obstruction is considered the most common presenting symptom of patients with SP.

For planning the surgical treatment, the authors found the classification system by C annady et al. to be very useful . In cases in which SPs are associated with SCC, radiation therapy appears to be an effective adjunctive procedure. Only one reported article on the metastasis of malignant SPs to the neck was found in the literature . Long-term follow up is recommended to detect recurrence, as disease can become extensive before it becomes symptomatic.

In conclusion, a high recurrence rate combined with the possibility of the multicentric origin of SPs should be managed early and treated aggressively. This case report highlights the importance of the en bloc surgical approach for the treatment of SPs, and present this rare report of neck metastasis associated with malignant sinonasal papillomas.