Role of ranulas in early diagnosis of Sjögren’s syndrome: A case report

Table 1 Laboratory parameters of the patient with primary Sjögren’s syndrome who had a ranula before

Laboratory parameters	Day 1 (March 1 on outpatient service)	After 2 wk of treatment	After 3 mo of treatment	After 6 mo of treatment	After one year and three mo of treatment
WBC (× 109/L)	3.42↓	3.97	4.25	4.79	4.25
Neutrophils, %	49.1↓	55.8	55.6	60.2	55.6
Lymphocytes, %	43.3↑	35.7	36.6	31.9	36.6
ESR (mm/h)	33↑	17	10	17	7
ALT (U/L)	23	18	17	13	24
AST (U/L)	31	23	27	24	29
Creatinine (μmol/L)	67	66	86	76	68
EGFR (mL/min)	110.5	112.5	81.7	94.2	107.8
ANA	1:320	-	-	1:320	1:320
Anti-dsDNA antibody	Negative	-	-	Negative	Negative
Anti-RNP/Sm antibody	Negative	-	-	Negative	Negative
Anti-Sm antibody	Negative	-	-	Negative	Negative
Anti-SSA antibody	Positive	-	-	Positive	Positive
Anti-SSB antibody	Positive	-	-	Positive	Positive
IgA (g/L)	1.60	-	-	1.66	1.57
IgG (g/L)	20.10↑	-	-	15.9↑	14.47
IgM (g/L)	1.15	-	-	1.00	1.08
C3 (g/L)	0.69↓	-	-	0.75↓	1.05
C4 (g/L)	0.18	-	-	0.19	0.26
RF (IU/mL)	441↑	-	-	165↑	46.86↑

WBC: White blood cells; ESR: Erythrocyte sedimentation rate; ALT: Alanine aminotransferase; AST: Aspartate aminotransferase; EGFR: Estimate glomerular filtration rate; ANA: Anti-nuclear antibody; dsDNA: Double-stranded DNA; RNP: Ribonucleoprotein; SSA: Sjögren’s syndrome–related antigen A; SSB: Sjögren’s syndrome–related antigen B; Ig: Immunoglobulin; C3: Complement component 3; C4: Complement component 4; RF: Rheumatoid factor.

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.