Submandibular and sublingual salivary gland involvement in adenoid cystic carcinoma

Sawant, Poonam Ramnath; Dhume, Manjeeta Mahesh Sinai; Spadigam, Anita; Dhupar, Anita

doi:10.4322/acr.2024.500

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Clinical Case Report and Review • Autops. Case Rep. 14 • 2024 • https://doi.org/10.4322/acr.2024.500 linkcopy

Submandibular and sublingual salivary gland involvement in adenoid cystic carcinoma

Authorship SCIMAGO INSTITUTIONS RANKINGS

Adenoid cystic carcinoma (AdCC) is a malignant salivary gland neoplasm that presents as an indolent but aggressive neoplasm. AdCC histogenesis is linked to the intercalated ducts of the salivary glands, equally affecting the major and minor glands. AdCC is associated with distant metastasis, most commonly to the lungs, and a high recurrence rate. AdCC accounts for 4.2% of all tumors. About 55% of all reported cases affect the submandibular gland, and around 50% of AdCC cases occur in the minor salivary glands. The present review describes a case of AdCC which presented a single nodular swelling on the right side involving the floor of the mouth. It also consolidates the histopathological profile of a case of AdCC with all the relevant histopathological features.

Keywords:
Carcinoma, Adenoid Cystic; Salivary Gland Diseases; Sublingual Gland; Submandibular Gland

Ref.	Age(y)/Gender	Site	Histopathology
Ref.	Age(y)/Gender	Site	Pattern	PN	PV	Other
⁷	58/F	Right SM region	Cribriform	-	-	-
⁸	57/F	Left FOM	Cribriform (Sublingual)	-	-	-
⁹	16/F	Right FOM	Cribriform +Tubular (Sublingual)	+	+	-
¹⁰	73/M	Right FOM	Cribriform + Solid (Sublingual)	-	-	-
¹¹	43/F	Left SL region	Cribriform	-	-	-
¹²	64/M	Right FOM	Cribriform (Sublingual)	-	-	-
PC	59/M	Right FOM	Cribriform + Tubular + Solid (Submandibular + Sublingual)	+ SH-HGT	+ SH-HGT	+ muscle

Invasion	Reference/ n cases	Prognosis
Perineural invasion	¹⁵/n=60	Margin status and perineural invasion predict disease-free survival or overall survival
	¹⁶/n=59	Although clinical T stage, gross nerve invasion, and nodal involvement before treatment were adverse prognostic factors, routine radiation of the skull base may reduce the bad prognostic significance of the perineural invasion
	¹⁷/n=33	Perineural invasion did not demonstrate a statistically significant association with survival.
	¹⁸/n=198	Perineural invasion was an adverse prognostic factor only when a major nerve was involved.
	¹⁹/n=30	Need to do a frozen section examination of the peripheral margins along with the adjacent nerve that is most likely involved.
	²² and ²⁰,	perineural or clinically named nerve invasion alarms for a worse prognosis
	²¹	Perineural involvement does not impact outcome.
Perivascular invasion	¹⁷/n=33	5 cases of Perivascular invasion (No mention of its significance)
Other tissue invasion	¹⁷/n=33	15 cases of muscle or tissue invasion (No mention of its significance)

Grading systems	Histopathological Grades of AdCC
Grading systems	Grade I	Grade II	Grade III
Spiro and Huvos³¹	Mostly Tubular or Cribriform, Occasionally Solid	Mixed with substantial Solid	Only solid
Perzin et al.³²/ Szanto et al.¹³	Predominantly Tubular, No solid	Predominantly Cribriform, <30% Solid	Solid component >30%
John and Chan³³	Tubular and cribriform together, without a solid pattern	Mostly cribriform, with less than 30% of solid pattern	Solid being the predominant subtype
van Weert et al.³⁴	No Solid component	Presence of Solid component (Any %)	-
Morita et al.⁵ Min A max-Minor Axis maximum	Min A max < 0.20 mm	Min A max >0.20 mm	-

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Hospital Universitário da Universidade de São Paulo Hospital Universitário da Universidade de São Paulo, Av. Prof. Lineu Prestes, 2565 - Cidade Universitária, 05508-000 - São Paulo - SP - Brasil, (16) 3307-2068, (16) 3307-2068 - São Paulo - SP - Brazil
E-mail: autopsy.hu@gmail.com

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[1] FOM= floor of mouth; PC= present case; PN= perineural; PV= perivascular; Ref = reference, SH-HGT= stroma hyalinization and high-grade transformation; SL= sublingual; SM= submandibular.