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LETTER TO EDITOR  
Year : 2018  |  Volume : 61  |  Issue : 4  |  Page : 632-634
Canalicular adenoma arising from the nasal septum in a child: First case report


Department of Pathology, Seth G. S. Medical College and KEM Hospital, Mumbai, Maharashtra, India

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Date of Web Publication10-Oct-2018
 

How to cite this article:
Ray M, Sathe P, Ghodke R, Suryavanshi M. Canalicular adenoma arising from the nasal septum in a child: First case report. Indian J Pathol Microbiol 2018;61:632-4

How to cite this URL:
Ray M, Sathe P, Ghodke R, Suryavanshi M. Canalicular adenoma arising from the nasal septum in a child: First case report. Indian J Pathol Microbiol [serial online] 2018 [cited 2018 Dec 15];61:632-4. Available from: http://www.ijpmonline.org/text.asp?2018/61/4/632/242963




Editor,

Minor salivary gland neoplasms unusually occur in the nasal cavity.[1] Canalicular adenoma (CA) is an uncommon benign neoplasm of the minor salivary glands, accounting for approximately 1% of all salivary gland tumors and 4%–6% of minor salivary gland tumors.[2],[3] A review of English literature revealed 525 cases of CA reported till date.[2],[4],[5] It is now recognized as a unique entity in the latest World Health Organization classification of salivary gland tumors, distinct from basal cell adenoma.[2],[3],[6]

An 11-year-old female child came with complaints of recurrent rhinitis and right-sided nasal obstruction for 2 months. On examination of the nose, there was a decrease in airblast in the right nostril, with a right-sided deviated nasal septum. A polypoidal mass was seen in the right nasal cavity, which did not bleed on touch. Ear and throat examination was within normal limits. A computed tomography (CT) scan of the paranasal sinuses revealed an S-shaped nasal septum and a soft-tissue density mass in the anterior part of the right nasal cavity with broad base toward the nasal septum, and extension into the anterior ethmoidal air cells [Figure 1]a and [Figure 1]b. There was no evidence of internal calcification/mucocele formation/hyperostosis/sinus obstruction/bone destruction/intracranial extension. The child was taken up for right functional endoscopic sinus surgery, where the mass was seen arising from the nasal septum. The excised specimen was sent for histopathological examination.
Figure 1: (a) (coronal view), (b) (axial view): Computed tomography scan showing soft density mass in anterior part of right nasal cavity with right-sided S-shaped deviated nasal septum. (c) Gross specimen, external surface– tan-brown, circumscribed, lobulated, polypoidal tumor. (d) Cut surface– homogenous, pale brown, with scattered slit-like spaces

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Grossly, the intact specimen was polypoidal, tan brown, measuring 2.5 cm × 1.8 cm × 0.3 cm. External surface was circumscribed and lobulated [Figure 1]c. On cut surface, the lesion was homogenous, pale brown with interspersed slit-like spaces containing blood [Figure 1]d.

Microscopic examination revealed a polypoidal, multilobulated lesion lined by pseudostratified, ciliated, columnar epithelium [Figure 2]a. The subepithelium showed proliferation of tumor cells arranged in closely packed acinar and glandular configurations [Figure 2]b, extending up to the base. Individual cells were cuboidal to columnar with eosinophilic cytoplasm and basally located, round-to-oval nuclei with stippled chromatin [Figure 2]c. The intervening stroma was edematous, had congested blood vessels, scattered lymphoplasmacytic inflammatory cells, and focal myxoid areas [Figure 2]d. There was no chondroid/cartilaginous elements/cellular atypia/mitosis/necrosis. Periodic acid–Schiff (PAS) and Alcian Blue (AB) stains showed focal positivity for mucoid material in the stroma [Figure 2]e and [Figure 2]f. On immunohistochemistry (IHC), the tumor cells were diffusely positive for the pan-cytokeratin [CK] and vimentin, focally positive for S-100 [Figure 2]g, [Figure 2]h, [Figure 2]i, and negative for synaptophysin, chromogranin, and alpha-smooth muscle actin (a-SMA). Based on these findings, a final diagnosis of CA of the nasal septum was made.
Figure 2: Microscopy: (a) Pseudostratified, ciliated, columnar epithelial lining (H and E, ×400). (b) Closely packed tumor cells in acinar and glandular patterns. Adjacent hypocellular stroma with embedded blood vessels (H and E, ×100). (c) Tumor cells showing pale, eosinophilic cytoplasm and basally located nuclei with stippled chromatin (H and E, ×400). (d) Myxoid stroma having mild, lymphoplasmacytic infiltrate (H and E, ×100). (e and f) PAS and AB stain–highlight mucoid material (H and E, ×400). (g) Diffuse pan-CK positivity (IHC, ×100). (h) Vimentin diffusely positive (IHC, ×100). (i) Focal S-100 positivity (IHC, ×100)

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CAs histologically, immunophenotypically, and ultrastructurally, most closely resemble luminal cells of the intercalated ducts, from which they are thought to arise.[2] The most common site is the upper lip (70%–90%), followed by buccal mucosa and palate, respectively. Rare cases have been reported in the lower lip and parotid.[2],[3] CAs have never been reported in the nasal cavity in English literature, as per our thorough review. Other salivary gland adenomas, such as basal cell adenomas and pleomorphic adenomas, although extremely rarely, are known to occur in the nasal septum.[7],[8] CA has only been reported in adults thusfar, with the most common age of presentation being above 60 years.[2],[3] It has never been reported in children.[2]

CAs are well circumscribed, maybe encapsulated, and are frequently lobulated/bosselated. Microscopically, the tumor cells are monomorphic, cuboidal to columnar, frequently double-layered or arranged in ductular configuration. The nuclear chromatin is stippled, may be focally hyperchromatic. Nucleoli are inconspicuous to focal and small. The surrounding stroma is hypocellular, contains many blood vessels, may be myxoid or fibrillar and show collagen deposition. On histochemical staining with PAS (with and without diastase), AB, and mucicarmine the stroma may be positive for mucoid material. Tumor cells shows positivity for pan-CK, vimentin, S-100, CD117, DOG1, SOX10 and may show a luminal or peripheral positivity for glial fibrillary acid protein at the tumor-connective tissue interface. Cells are negative for p63 and myoepithelial markers including, desmin, a-SMA, muscle specific actin, smooth muscle myosin heavy chain, and calponin. Our case had similar gross and microscopic features. In view of the predominant acinar pattern and stippled chromatin, our differentials included CA, basal cell adenoma-trabecular type, pleomorphic adenoma, and neuroendocrine neoplasm. However, on account of focal PAS and AB stain positivity [absent in basal cell adenoma], and IHC revealing diffuse pan-CK and vimentin positivity, focal S-100 positivity, and negativity for a-SMA (positive in basal cell adenoma and pleomorphic adenoma), synaptophysin, and chromogranin (ruling out neuroendocrine neoplasm), a final diagnosis of CA of the nasal septum was rendered.

CAs are also rarely known to be multifocal, with nodules presenting distinctly away from the main tumor, or microscopic evidence of satellite nodules. CAs have no malignant potential.[3]

Radiologically, CT and MRI imaging are not diagnostically specific for CA.[3] However, they may be of some preoperative diagnostic help, by including CA in differentials of lesions.

Treatment options include biopsy, enucleation, and excision. Since local recurrences and multifocality are known to occur, the definitive cure may be assured by surgical excision inclusive of a surrounding narrow rim of uninvolved parent tissue. Patients should be kept on follow-up over the near future. Our case had involvement of the base of excision and hence has been advised follow-up. She is presently asymptomatic and is doing well, 10 months postoperatively.

This is the first case of CA to be reported in a child and also the first case involving the nasal septum. Thus, CAs should be kept in mind for a nasal cavity mass, even in the pediatric age group, and appropriate management should be undertaken in light of its known complications.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
McKenna RJ. Tumors of the major and minor salivary glands. CA Cancer J Clin 1984;34:24-39.  Back to cited text no. 1
    
2.
Thompson LD, Bauer JL, Chiosea S, McHugh JB, Seethala RR, Miettinen M, et al. Canalicular adenoma: A clinicopathologic and immunohistochemical analysis of 67 cases with a review of the literature. Head Neck Pathol 2015;9:181-95.  Back to cited text no. 2
    
3.
Azevedo LR, Dos Santos JN, De Lima AA, Machado MA, Grégio AM. Canalicular adenoma presenting as an asymptomatic swelling of the upper lip: A case report. J Contemp Dent Pract 2008;9:91-7.  Back to cited text no. 3
    
4.
Bajpai M. Canalicular adenoma arising in buccal mucosa. J Coll Physicians Surg Pak 2016;26:946.  Back to cited text no. 4
    
5.
Ortega RM, Bufalino A, Almeida LY, Navarro CM, Travassos DC, Ferrisse TM, et al. Synchronous polymorphous adenocarcinoma and canalicular adenoma on the upper lip: An unusual presentation and immunohistochemical analysis. Head Neck Pathol 2018;12:145-9.  Back to cited text no. 5
    
6.
El-Naggar AK, Chan JK, Grandis JR, Takata T, Slootweg PJ, editors. WHO Classification of Head and Neck Tumours. 4th ed. Lyon: IARC; 2017. p. 160.  Back to cited text no. 6
    
7.
Wang Q, Chen H, Wang S. Basal cell adenoma of nasal septum: Report of a case and review of literature. Int J Clin Exp Pathol 2015;8:2176-9.  Back to cited text no. 7
    
8.
Sciandra D, Dispenza F, Porcasi R, Kulamarva G, Saraniti C. Pleomorphic adenoma of the lateral nasal wall: Case report. Acta Otorhinolaryngol Ital 2008;28:150-3.  Back to cited text no. 8
    

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Correspondence Address:
Manjusha Ray
Department of Pathology, Seth G. S. Medical College and KEM Hospital, Acharya Donde Marg, Parel, Mumbai - 400 012, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_137_18

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