Department of Oral Diagnostic Sciences, King AbdulAziz University - Faculty of Dentistry, Jeddah, Saudi Arabia.
Department of Periodontolgy, King AbdulAziz University - Faculty of Dentistry, Jeddah, Saudi Arabia.
Am J Case Rep. 2022 Jun 13;23:e936192. doi: 10.12659/AJCR.936192.
BACKGROUND Amyloidosis is a clinical condition characterized by the extracellular deposition of insoluble, abnormal amyloid fibrils in various body tissues. It is generally categorized into 2 forms - localized and systemic - with a wide range of signs and symptoms. This case report discusses the localized amyloidosis involvement of the oral cavity and its treatment. CASE REPORT A 65-year-old woman presented to the oral medicine clinic reporting painless tongue enlargement, which has been slowly progressing over several years, leading to difficulty in tongue movement, eating, and swallowing. Extra-oral examination showed a prominent lower lip with rubbery consistency. Intra-oral examination revealed a significantly enlarged tongue almost filling the whole oral cavity with dental indentations evident on all tongue surfaces and multiple, deep ulcerative craters of various sizes ranging from 2 to 5 mm in diameter. Histopathological examination under light microscope using hematoxylin and eosinophil and Congo red stain were diagnostic for amyloidosis. Further investigation with the Rheumatology Department, including renal and liver function tests, as well as echocardiography, were conducted and ruled out systemic involvement of other body organs. The patient was treated with weekly intra-lesional triamcinolone injections, with significant improvement. CONCLUSIONS We report a rare case of localized amyloidosis presenting as macroglossia. Although the most effective management in tongue amyloidosis is surgical resection, conservative management in cases of localized oral amyloidosis presenting as macroglossia with weekly intra-lesional triamcinolone injections can be an effective approach, providing patients with better quality of life. Future studies exploring treatment modalities for similar cases with limited oral involvement are warranted.
淀粉样变性是一种临床病症,其特征是细胞外沉积不可溶的、异常的淀粉样纤维,发生于各种身体组织中。淀粉样变性通常分为局部型和系统性,具有广泛的体征和症状。本病例报告讨论了口腔局部淀粉样变性及其治疗方法。
一名 65 岁女性因无痛性舌体增大到口腔医学门诊就诊,该症状已缓慢进展数年,导致舌运动、进食和吞咽困难。体格检查发现下唇明显肿大,质地如橡皮。口腔检查显示舌体显著增大,几乎填满整个口腔,所有舌面均可见牙印,且有多个大小不一的深溃疡凹陷,直径 2 至 5 毫米不等。光镜下组织病理学检查,包括苏木精和伊红及刚果红染色,诊断为淀粉样变性。与风湿科合作进行了进一步检查,包括肾功能和肝功能检查以及超声心动图检查,排除了其他身体器官的系统性受累。每周对患者进行腔内曲安奈德注射治疗,病情明显改善。
我们报告了一例罕见的局部型淀粉样变性表现为巨舌症的病例。虽然舌淀粉样变性最有效的治疗方法是手术切除,但对于表现为巨舌症的局部口腔淀粉样变性,腔内每周曲安奈德注射的保守治疗是一种有效的方法,可以提高患者的生活质量。需要进一步研究探索类似病例、口腔受累有限的治疗方法。