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An interesting case of oral inverted ductal papilloma Natasha Berridge Mahesh Kumar Dental Update 2024 43:10, 707-709.
Authors
NatashaBerridge
BSc(Hons), BDS, BM, MFDS(Eng), MRCS(Eng)
Department of Oral Surgery & Orthodontics, The Hillingdon Hospital, Pield Heath Road, Uxbridge, Middlesex UB8 3NN, UK (natasha.berridge@googlemail.com)
Ductal papillomas are rare benign papillary tumours of the minor salivary glands. Previously they have been classified into three subtypes: inverted ductal papilloma (IDP), sialadenoma papilliferum and intraductal papilloma. The oral inverted ductal papilloma is the least common of these lesions, with 48 cases reported to date in a search of the English-language literature. We describe an interesting case of oral IDP and highlight the pertinent clinical and histopathological features. Importantly, unlike the histologically similar inverted papilloma (IP) of the nose and paranasal sinuses, oral IDP is a completely benign lesion and is not associated with malignant change.
CPD/Clinical Relevance: This case report aims to raise awareness of the lesion, oral inverted ductal papilloma (IDP). We outline the clinical features and unique histopathology of oral IDP so that clinicians may correctly diagnose such a lesion and implement appropriate treatment.
Article
Ductal papillomas are rare, benign, papillary tumours of the minor salivary glands. They represent adenomas with unique histopathological features that arise from the excretory ducts of the salivary glands. The World Health Organization (WHO)1 classifies ductal papillomas into three sub-types: inverted ductal papilloma (IDP), sialadenoma papilliferum and intraductal papilloma.
The oral IDP, first described by White et al (1982),2 is the least common lesion of the three subtypes, with a reported 48 cases to date in a search of the English-language literature.2,3,4,5,6,7,8,9,10,11,12,13,14 We describe an interesting case of IDP occurring in the buccal mucosa of an elderly woman, including the histopathological findings and pertinent clinical features.
Case report
A 72-year-old woman was referred to the Department of Oral and Maxillofacial Surgery at Hillingdon Hospital by her GDP regarding a ‘lump’ in the right buccal mucosa of the lower premolar area of 3 years' duration. Aside from a gradual increase in size, the lump had been otherwise asymptomatic.
Her medical history of note included essential hypertension and excisional biopsy of a palatal polyp ten years previously. She was taking bendroflumethiazide and simvastatin, and had no known drug allergies. She had smoked 4–5 cigarettes/day for the past 20 years and consumed 14 units of alcohol/week.
On examination, there was a 1.5 cm x 1 cm lesion that was raised, mobile and soft with normal overlying oral mucosa; and a central crater-like defect with whitish rolled edges (Figure 1). The lesion was not painful and there was no associated cervical lymphadenopathy.
Figure 1 Pre-operative view showing the lesion on the buccal mucosa. There is a mobile and soft submucosal swelling with a moderate-sized punctum on the mucosal surface.
An urgent incisional biopsy was performed under local anaesthesia. Microscopic examination revealed a diagnosis of IDP. The squamous mucosa, including some deep salivary gland tissue, consisted of a large collection of moulded lobules composed of biphasic basaloid and clear cells, with both large and small cystic spaces. There was no atypia and mitotic figures were only rarely seen.
Following the biopsy and diagnosis of IDP, formal wide local excision of the lesion under local anaesthesia was performed. The resultant microscopic report stated that there was an exoendophytic squamous epithelial proliferation comprising complex invaginations of bland squamous epithelium with admixed mucus cells and slit-like cystic spaces lined by columnar epithelial cells. There were no findings suggestive of a malignant tumour (Figures 2 and 3).
Figure 2 At low power (H&E stain, x20) the tumour shows an inverted growth pattern composed of complex invaginations of epithelium.Figure 3 At high power (H&E stain, x400) the tumour shows well-differentiated squamous epithelium with occasional mucous cells and a lining of columnar cells.
The post-operative wound healed well. However, the patient reported some numbness of the right lower lip. On review, 4 months later, the right lower lip numbness had significantly resolved, leaving only a small area of sensory loss in the midline.
At 18 months review, there was no sign of recurrence clinically and the post-operative numbness was still limited to a small portion of the central lower lip. There had been no signs of recurrence after 2 years follow-up and the lower lip anaesthesia had resolved.
Discussion
Oral IDP is a distinct clinical lesion that is histologically similar to the more common inverted papilloma (IP) of the nasal cavity, paranasal sinuses, urinary tract, cervix and lacrimal sac. A completely benign lesion, oral IDP is not associated with malignant change, unlike IP of the nasal cavity and paranasal sinuses. It has been reported that these nasal and paranasal papillomas are destructive, frequently recur, and are associated with squamous cell carcinoma in 10–15% of cases.12
According to the literature, the most common locations for IDP within the oral cavity were the lips and buccal mucosa, followed by the palate, mandibular vestibule and floor of the mouth. Patients were aged between 28–77 years, with a peak in the sixth and seventh decade, and a mean age of 51 years. There was a female-to-male preponderance of 2.2 to 1.
Clinically, all documented IDP lesions have presented with a submucosal nodule, with or without a central punctum, and have ranged in size from 0.5–1.5 cm. The lesions appear to be connected to the surface, but are otherwise moveable. The clinical duration of the lesions varied from several months to 29 years. This may present a challenge for the clinician, as several other oral mucosal lesions bear a striking resemblance to oral IDP clinically.
Differential diagnosis should include benign lesions, such as mucocele, lipoma, fibroma and infection (eg syphilitic chancre); the more sinister malignant tumours of the salivary glands; and oral squamous cell carcinoma. To date, all cases of oral IDP have been treated by complete surgical excision, and there have been no cases of malignant transformation or recurrence reported.
Histologically, IDP resembles IP of the nasal cavity and appears to be the result of a process of proliferation and squamous metaplasia of a minor salivary gland excretory duct. The tumour shows a characteristic endophytic growth pattern and is composed of bulbous papillary projections of basaloid and squamous cells dilating and filling the superficial portion of the excretory duct. Unlike IP of the nasal cavity and paranasal sinuses, keratinization is not a feature of ductal papillomas. IDPs have unique, readily identifiable features that distinguish them from other lesions.
Conclusion
Oral IDP is a rare tumour of the minor salivary glands. An accurate clinical examination and biopsy for histopathological analysis represent the baseline work-up that is needed to diagnose and surgically treat oral IDP correctly. It is important to raise awareness of this benign clinical entity amongst clinicians of the head and neck region, to ensure appropriate follow-up, including the recommended complete surgical excision.
