Kimura Taku, Shimura Takumi, Kato Yasushi, Yanagawa-Matsuda Aya, Kawamura Kazuhiro, Tanei Zen-Ichi, Tanaka Shinya, Sakata Ken-Ichiro
Department of Oral Diagnosis and Medicine, Hokkaido University, Sapporo, JPN.
Department of Vascular Biology and Molecular Pathology, Faculty and Graduate School of Dental Medicine, Hokkaido University, Sapporo, JPN.
Cureus. 2025 Aug 27;17(8):e91117. doi: 10.7759/cureus.91117. eCollection 2025 Aug.
Adenomatoid hyperplasia (AH) of the minor salivary glands is a rare, benign hyperplastic lesion that typically arises in the oral mucosa, most commonly in the hard or soft palate. AH generally presents as a painless mass covered with normal mucosa. AH can mimic the clinical course of malignant salivary gland tumors, making diagnosis challenging. This series presents two cases of AH arising in unusual sites, the buccal mucosa and sublingual caruncle, which were initially suspected of being malignant tumors and were evaluated using multimodal imaging. Patient 1, a 76-year-old female, presented with a buccal mass of 6 × 8 mm in size that increased in size over time and was eventually diagnosed with AH with sialadenitis. Patient 2, an 85-year-old male, presented with an indurative mass of 4 × 4 mm in size near the sublingual caruncle, later diagnosed with AH along with sialadenitis. Both patients underwent excisional biopsy with a safety margin comparable to that for malignant tumors. Each patient is now being monitored regularly without recurrence. Our patients underwent multiple imaging modalities prior to the surgery; however, they underscored the limited utility, especially in evaluating small lesions. Therefore, histopathological examination remains crucial for its definitive diagnosis. AH can present in unusual locations and mimic malignancy during its clinical course, especially when accompanied by inflammation, emphasizing the importance of its differential diagnoses. Through our case series, we highlighted that excisional biopsy is an effective strategy, and histopathological examination is essential for accurate diagnosis.
小涎腺腺瘤样增生（AH）是一种罕见的良性增生性病变，通常发生于口腔黏膜，最常见于硬腭或软腭。AH一般表现为覆盖正常黏膜的无痛性肿块。AH可模仿涎腺恶性肿瘤的临床病程，给诊断带来挑战。本系列报告了两例发生于不寻常部位（颊黏膜和舌下肉阜）的AH病例，最初怀疑为恶性肿瘤，并采用多模态成像进行评估。患者1为一名76岁女性，表现为大小为6×8mm的颊部肿块，肿块随时间增大，最终诊断为AH伴涎腺炎。患者2为一名85岁男性，在舌下肉阜附近出现大小为4×4mm的硬结性肿块，后来诊断为AH伴涎腺炎。两名患者均接受了切除活检，切缘安全范围与恶性肿瘤相当。目前两名患者均在定期监测中，无复发。我们的患者在手术前接受了多种成像检查；然而，这些检查的效用有限，尤其是在评估小病变时。因此，组织病理学检查对于明确诊断仍然至关重要。AH可出现在不寻常的部位，在临床病程中模仿恶性肿瘤，尤其是伴有炎症时，这凸显了其鉴别诊断的重要性。通过我们的病例系列，我们强调切除活检是一种有效的策略，组织病理学检查对于准确诊断至关重要。
