Chen Na, Zeng Da-Shun, Su Yu-Tong
Department of Endocrine and Metabolic Diseases, Shanghai Institute of Endocrine and Metabolic Diseases, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China.
Department of Oral Surgery, The Third Affiliated Hospital of Wenzhou Medical University, Wenzhou 325200, Zhejiang Province, China.
World J Clin Cases. 2021 Jul 16;9(20):5701-5708. doi: 10.12998/wjcc.v9.i20.5701.
Although the presentations of Sjögren's syndrome (SS) are variable, ranging from mild dryness to wider systemic involvement, ranulas as early clinical signs were scarcely reported. Here, we present an adult patient with SS, who developed a unilateral simple ranula and was diagnosed primary SS 3 years later. We also provide a review of cases of SS and ranulas from 1980 to 2020.
A 22-year-old girl was found to have a left painless floor-of-mouth lesion 3 years ago, without obvious trauma or inducement. The diagnosis of a unilateral (left) simple ranula was made, and the ranula was surgically treated. Within 3 years after the ranula surgery, she developed acute lymphadenectasis in unilateral parotid twice without inducement, and ultrasonic examination revealed diffuse lesions in bilateral parotids and submandibular glands, which strongly suggested SS. Serologic tests and the unstimulated whole saliva flow rate confirmed the SS diagnosis.
Our study underlines that ranulas are early clinical signs of SS. As early diagnosis and early intervention of SS are important to obtain better outcomes, our findings underline the need for histopathological test after sublingual adenectomy and imaging detection of exocrine glands for the patients with ranulas.
尽管干燥综合征(SS)的表现多种多样,从轻度干燥到更广泛的全身受累,但鲜有报道称舌下囊肿是其早期临床症状。在此,我们报告一例成年SS患者,该患者出现单侧单纯性舌下囊肿,并在3年后被诊断为原发性SS。我们还对1980年至2020年SS合并舌下囊肿的病例进行了综述。
一名22岁女孩3年前被发现左侧口底有一无痛性病变,无明显外伤或诱因。诊断为单侧(左侧)单纯性舌下囊肿,并对该舌下囊肿进行了手术治疗。在舌下囊肿手术后3年内,她两次无诱因出现单侧腮腺急性淋巴结肿大,超声检查显示双侧腮腺和颌下腺弥漫性病变,强烈提示SS。血清学检查和非刺激性全唾液流速测定证实了SS诊断。
我们的研究强调舌下囊肿是SS的早期临床症状。由于SS的早期诊断和早期干预对于获得更好的治疗效果很重要,我们的研究结果强调对于舌下囊肿患者,在舌下腺切除术后需要进行组织病理学检查,并对外分泌腺进行影像学检测。