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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 12  |  Issue : 1  |  Page : 57-60

Intraoral ductal papilloma of the lower lip in a 6-year-old patient


Department of Oral Pathology, SRM Dental College, Chennai, Tamil Nadu, India

Date of Submission03-Jan-2021
Date of Decision16-Feb-2021
Date of Acceptance19-Feb-2021
Date of Web Publication30-Mar-2021

Correspondence Address:
Dr. A E Nagarathinam
SRM Dental College, Ramapuram, Chennai, Tamil Nadu
India
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DOI: 10.4103/srmjrds.srmjrds_2_21

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  Abstract 

Ductal papilloma is a type of rare salivary gland tumor of minor salivary glands and commonly seen in the lower lip and buccal vestibule. Ductal papilloma usually presents in the form of three histopathological variants, namely sialedenoma papilliferum, intraductal papilloma, and inverted ductal papilloma. The etiopathogenesis of a ductal papilloma remains unclear, but some authors suggest the association with human papilloma virus. Diagnosis of intraoral ductal papilloma predominantly depends on the histopathological features; however, clinical differential may include benign lesions such as mucocele, lipoma, and fibroma. Considering the benign and nonaggressive nature of such tumors, histopathology alone is suitable for the diagnosis. However, immunohistochemistry may be used in cases of aggressive growth or in malignant counterpart. In this case report, we report the rare case of intraoral ductal papilloma of the lower lip mimicking a mucocele in a 6-year-old female patient.

Keywords: Benign tumours, ductal papilloma, pediatric, salivary glands


How to cite this article:
Swaathi R, Sankari U, Swetha I S, Pokkali T, Murali S, Nagarathinam A E. Intraoral ductal papilloma of the lower lip in a 6-year-old patient. SRM J Res Dent Sci 2021;12:57-60

How to cite this URL:
Swaathi R, Sankari U, Swetha I S, Pokkali T, Murali S, Nagarathinam A E. Intraoral ductal papilloma of the lower lip in a 6-year-old patient. SRM J Res Dent Sci [serial online] 2021 [cited 2021 Jun 25];12:57-60. Available from: https://www.srmjrds.in/text.asp?2021/12/1/57/312476


  Introduction Top


Ductal papilloma is a term usually used to equate with a group of three rare minor salivary gland tumors which shows peculiar papillomatous features. They are sialedenoma papilliferum, intraductal papilloma, and inverted ductal papilloma.[1] They are common in the minor salivary glands than in the major salivary glands, and inverted oral ductal papilloma is the least common lesion and reported only in minor salivary glands among adults. Sialadenoma papilliferum demonstrates the features with both endophytic and exophytic squamous features. Intraductal papilloma is often muddled with papillary cystadenoma which is also a salivary gland lesion. The etiopathogenesis of Intraductal papilloma (IDP) still remains unclear, but few authors relate it to human papilloma virus.[2] The most common site of involvement is the lower lip followed by the buccal vestibular mucosa. Lesions such mucocele, lipoma, fibroma, and infection are the usual suggestive differential diagnosis for oral ductal papilloma. The exact incidence of the lesions is not available due to the usage of inconsistent terminology used in describing the lesion as “papillary cystadenoma” instead of the term “intraductal papilloma.”[3],[4] The age range was from 28 to 77 years, with a mean age of around 56 years with no obvious sex predilection.[5] Male predilection is seen in Sialedenoma Papilliferum.[1] In this case report, we report a case of intraoral ductal papilloma of the lower lip mimicking a mucocele in a 6-year-old female patient.


  Case Report Top


A 6-year-old young female reported to the outpatient ward of oral medicine department with the complaint of painless swelling in the right side lower lip for the past 1 month and also reports difficulty in chewing. The patient gives a history of painless swelling present for the past 1 month; however, the swelling was recurrent in nature during the past 1 year. The patient also gives a history of surgical removal of swelling in the same site 1 year ago and reports uneventful healing during that period. History also reveals that the patient is a chronic lip biter. The current swelling is present for the past 1 month.

On clinical intraoral examination, the swelling was small, round, movable, well circumscribed, elevated in relation to the normal mucosa, was soft to mildly firm in consistency and measuring roughly around 6 mm in diameter and nontender on palpation [Figure 1]. Extraoral examination did not reveal any abnormal finding, no palpable lymph nodes, medical history was noncontributory. An excisional biopsy was planned and done; the surgical specimen removed was well defined was easily removed from the surrounding tissue. Surgical closure was done, 1-week postoperative healing was uneventful. Clinical differential of fibroma and mucocele was considered.
Figure 1: Clinical presentation of the swelling on the lower lip (right side)

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The surgically excised tissue was fixed in 10% formalin and sent for the histopathological examination. Hematoxylin and eosin staining was done. Microscopically, the section shows hyperplastic stratified squamous epithelium. The connective tissue showed eosinophilic areas suggestive of mucin which was surrounded by intense inflammatory infiltrate composed of neutrophils, macrophages, and lymphocytes. On deeper sectioning, areas showed ductal epithelium which was hyperplastic and proliferating into the lumen, exhibiting squamous metaplasia. Salivary gland acini and few blood vessels were seen. Final diagnosis was a intra Oral Salivary Ductal Papilloma [Figure 2] and [Figure 3].
Figure 2: Low power (×4 magnification) shows superficial stratified squamous epithelium which is mildly hyperplastic. The connective tissue showed number budding capillaries and eosinophilic areas suggestive of mucin which was surrounded by intense inflammatory infiltrate composed of neutrophils, macrophages, and lymphocytes

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Figure 3: High power (×40 Magnification) in deeper areas shows an area of ductal epithelium which was hyperplastic and proliferating into the lumen, also exhibiting squamous metaplasia

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  Discussion Top


Salivary gland neoplasms are usually occasional, and ductal papilloma is one among the rare benign papillary lesions arising from the salivary ductal system.[1],[6] Intraductal papilloma is the rarest among the ductal papilloma of salivary glands. They usually have many similarities such as nonaggressive behavior, common in minor salivary glands, etc. White et al. in 1982 first described inverted ductal papilloma of minor salivary gland occurring in the in the floor of the mouth, lower lip, cheek, and soft palate.[7] The term inverted papilloma was coined owing to the resemblance of the similar type of lesion arising in the urinary bladder, nasal cavity, or paranasal sinuses. Lesions with similar features have also be observed within the pelvis, tear sac, cervix, and posterior pharyngeal wall. Sialadenoma Papilliferum, first described by Abrams and Finck in 1969, shows both exophytic and endophytic squamous feature, the lesion shows male predilection. Clinically, it resembles squamous papilloma. The exact incidence of intraductal papilloma remains unknown.[2],[3] It is usually difficult to determine the incidence of salivary ductal gland tumors due to the usage of different terminologies in the literature, and only <50 cases of oral intraductal papilloma in minor salivary glands have been reported. The term “Papillary cystadenoma” is commonly used rather than “Intraductal papilloma.”[3] They are also called as “Simple Ductal Papilloma.” Size of the swelling usually ranges between 1 and 1.5 cm.[8] Gender distribution has usually remained even.[8] It is usually asymptomatic. Intraductal papilloma is usually common in breasts. In the oral cavity, it commonly occurs in the lower lip, whereas other sites include upper lip, buccal mucosa, and palate. Their occurrence is very rare in the major salivary gland. In case of occurring in major salivary glands, they are common in parotid.[1],[3] Other diagnostic modalities such as radiography and fine-needle aspiration do not show greater significance in the preoperative diagnosis. Histologically, intraductal papilloma shows ductal epithelium with papillomatous proliferation with a thin core of fibrovascular tissue, the papillary lesion is usually seen occupying the ductal space adjacent to salivary ductal areas. The lesion may present as a dilated unicystic structure located below or deep to the mucosal surface.[3],[8] The epithelial lining in the intraductal papilloma is usually lined by a single or double row of cuboidal or columnar epithelium characterized with numerous papillary projections into the cystic lumen. Ductal ectasia and/or ductal hyperplasia may be seen following the blockage of salivary ducts.[3] The difference between intraductal and inverted ductal papilloma is that the latter expands into to the connective tissue while the former is well-circumscribed unicystic lesion. As a result of epithelial proliferation, inverted ductal papilloma seems to be arising from the excretory ducts closer to the mucosal surface while intraductal papilloma arises from the excretory ducts at a deeper level.[3],[9] Immunohistochemically, the origin of the tumors cells has been identified to be from ductal luminal cells, ductal epithelium, and excretory ductal epithelium. Strong positivity of Keratin and Epithelial Membrance Antigen was seen in cells facing the lumina and mildly reactive for lactoferrin and S-100.[9] Other immunohistochemical studies revealed reactivity with cytokeratins, epithelial membrane antigen, carcinoembryonic antigen, and lysozyme among the proliferating epithelial cells. Before diagnosis, it is always essential to rule out the tumors. Differential diagnosis of intraductal papilloma includes lesions such as inverted ductal papilloma, sialadenoma papilliferum, Warthin's tumor, papillary cystadenoma, polymorphous low-grade carcinoma, papillary cystic variant of acinic cell carcinoma, and papillary cystadenocarcinoma.[3] Papillary cystadenoma is often observed with benign neoplastic papillary proliferation of the salivary gland duct epithelium in the form of multiple epithelium-lined cystic structures.[8] Papillary cystadenocarcinoma (malignant counterpart of cystadenoma) is characterized by multilocular, papillary, and cystic lesion with aggressive infiltrative growth. Despite being benign, it is often necessary to treat the tumor because there is always a chance of it turning into malignancy. Salivary duct carcinoma frequently shows intraductal epithelial proliferation. Salivary ductal carcinoma is often presented with distinct features like comedonecrosis, typical cribriform and Roman-bridge structures, and prominent cellular atypia distinguishing it from other benign lesions such as ductal papilloma.[10] Ductal papilloma needs to be differentiated from mucoepidermoid carcinoma as both lesions present with epidermoid and mucous cells. Although the diagnosis may have varied differential diagnosis, the treatment is usually complete excision along with long-term follow-ups, and the recurrence is rare.[4] In our case, the intraoral ductal papilloma was seen in a 6-year-old individual as opposed to more occurrence in adults. The recurrence rate and the lip biting habit of the patient made it appear like a mucocele which is a feature that is somewhat different from other cases. This case report could further help practitioners help distinguish mucocele from ductal papilloma and also reveals that ductal papilloma can be seen in children as well.


  Conclusion Top


The rarity of occurrence of a benign salivary gland tumor like intraoral ductal papilloma in a young 6-year-old female pediatric patient adds key to the significance of this case report. Although preliminary histopathological examination revealed areas suggestive of a mucocele like mucinous areas, deeper examination showed an area suggestive of ductal papilloma. If doubtful of a ductal papilloma of relatively smaller size as in this case, it may be safe to excise under local anesthesia itself. Excision of the lesion is the primary mode of treatment. It does not usually recur after complete removal.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Rajendran R. Shafer's Textbook of Oral Pathology. India: Elsevier; 2009.  Back to cited text no. 1
    
2.
Infante-Cossio P, Gonzalo DH, Hernandez-Gutierrez J, Borrero-Martin JJ. Oral inverted ductal papilloma associated with condyloma acuminata and HPV in an HIV+patient. Int J Oral Maxillofac Surg 2008;37:1159-61.  Back to cited text no. 2
    
3.
Brannon RB, Sciubba JJ, Giulani M. Ductal papillomas of salivary gland origin: A report of 19 cases and a review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:68-77.  Back to cited text no. 3
    
4.
Ramaswamy P, Khaitan T, Anuradha A, Kumar BP, Sudhakar S. Intraductal papilloma: Atypical presentation. Case Rep Dent 2013;2013:652728.  Back to cited text no. 4
    
5.
Cabov T, Macan D, Manojlović S, Ozegović M, Spicek J, Luksić I. Oral inverted ductal papilloma. Br J Oral Maxillofac Surg 2004;42:75-7.  Back to cited text no. 5
    
6.
Jansisyanont P, Blanchaert RH Jr., Ord RA. Intraoral minor salivary gland neoplasm: A single institution experience of 80 cases. Int J Oral Maxillofac Surg 2002;31:257-61.  Back to cited text no. 6
    
7.
White DK, Miller AS, McDaniel RK, Rothman BN. Inverted ductal papilloma: A distinctive lesion of minor salivary gland. Cancer 1982;49:519-24.  Back to cited text no. 7
    
8.
Ellis GL, Auclair PL, Gnepp DR. Surgical Pathology of the Salivary Glands: Volume 25 in the Major Problems in Pathology Series. Philadelphia: WB Saunders Company; 1991.  Back to cited text no. 8
    
9.
Hara H, Oyama T, Omori K, Misawa T, Kasai H, Kimura M, et al. Fine needle aspiration cytology of an intraductal papilloma originating in a sublingual gland. A case report. Acta Cytol 1999;43:457-63.  Back to cited text no. 9
    
10.
Nagao T, Sugano I, Matsuzaki O, Hara H, Kondo Y, Nagao K. Intraductal papillary tumors of the major salivary glands: Case reports of benign and malignant variants. Arch Pathol Lab Med 2000;124:291-5.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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