Table 2.

Review of primary Sjögren’s syndrome patients associated with ranulas

Ref.	Country		No. of cases	Age/gender	First visit department	Type of ranula	Sicca symptoms	Extra-landular symp-toms	Time interval between pSS diagnosis and detection of ranula	Antibody positivity	Treatment of ranula	Treatment of pSS	Outcome
Katayama et al[12], 1993	Japan		2	33, F; 34, F	Patient 1: dermatology department Patient 2: NA	Unilateral	Patient 1: oral dryness, caries, alopecia, and pruritic skin rash Patient 2: oral dryness	NA	Patient 1: after SSPatient 2: 7 yr after SS	ANA, SSA	Surgically treated	NA	NA
Pinheiro et al[13], 2017	Brazil		1	37, F	Clinic of the Dental School	Unilateral	Irritation of the eyes, enlargement of the parotid glands, and dry mouth	NA	1 yr after SS	ANA, SSA and SSB	Surgically treated	Prednisone, methotrexate, chloroquine diphosphate, and artificial tears and saliva	Improving all signs and symptoms
Means et al[8], 2017	United States		1	10, M	Pediatric Otolaryngology clinic	Bilateral	Bilateral recurrent parotitis, dry eyes, dental caries	NA	4 yr before SS	ANA, SSA and SSB	Transoral excision and marsupial-zation	Routine follow-up	NA
Lieberman et al[9], 2018	United States	2		12, F; 8, F	Patient 1: Dental Department; Patient 2: Otolaryngology	Patient 1: Bilateral; Patient 2: Unilateral	Patient 1: Recurrent parotid gland discomfort; Patient 2: No	Patient 1: Severe joint pain; Patient 2: no	Patient 1: 7 yr before SS; Patient 2: Simultaneous	Patient 1: SS-APatient 2: ANA, SSA, SSB and RF	Patient 1: surgically treated; Patient 2: Surgically treated	NA	Patient 1: NAPatient 2: Developed significant dry eyes
Sato et al[11], 2019	Japan	3		66, F; 30, F; 26, F	Department of Oral Medicine	NA	NA	NA	Simultaneous	SSA, SSB	Patients 1 and 2: Surgically treated; Patient 3: Naturally resolved	NA	NA
Takagi et al[10], 2020	Japan	7		12, M33, F41, F43, F46, F48, F51, F	NA	Patient 1: Bilateral, plunging; Patients 2-7: NA, simple	NA	NA	Patient 1: Simultaneous; Patients 2-7: NA, but before SS	Patient 1: ANA, SSA, SSB and RF; Patients 2-7: NA	Patient 1: Open fenestration; Patients 2-7: NA	Patient 1: Mizoribine and cortices-teroid medication; Patients 2-7: NA	Patient 1: Developed parotitis after 9 mo; Patients 2-7: NA
Present case	China	1		25, F	Oral surgery	Unilateral, simple	Ranula and recurrent parotitis	No	3 yr before SS	ANA, SSA and RF	Surgically treated	Hydroxychloroquine sulfate and total glycosides of paeony root capsule	No symptoms

M: Male; F: Female; pSS: Primary Sjögren’s syndrome; ANA: Antinuclear antibodies; SSA: Sjögren’s syndrome–related antigen A; SSB: Sjögren’s syndrome–related antigen B; RF: Rheumatoid factor; NA: Not available.