Ruba Mshref, Ahmad Alkheder, Nasser Alia, Rawad Salama, Mariana Naief Sharaf Aldeen, Raouf Salem Seif Eddin
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A literature review of 16 published cases (including ours) demonstrated a slight female predominance (10/16), variable symptom profiles (painless swelling to masticatory dysfunction), and diverse management strategies. Minimally-invasive Btx A achieved favorable outcomes in 6/16 cases, with lower doses (25 U) showing efficacy comparable with higher doses. Surgical intervention, although effective, was associated with recurrence in 1 case. Diagnostic reliance on imaging (CT/magnetic resonance imaging) and histology (when performed) confirmed hypertrophy without malignancy. While bruxism and stress were implicated in some cases, 7/16 lacked identifiable triggers, suggesting multifactorial etiology. Long-term follow-up data remain limited, underscoring the need for vigilant monitoring. This case reinforces Btx A as a primary therapeutic option and highlights the importance of individualized management in IUTMH. 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Diagnostic reliance on imaging (CT/magnetic resonance imaging) and histology (when performed) confirmed hypertrophy without malignancy. While bruxism and stress were implicated in some cases, 7/16 lacked identifiable triggers, suggesting multifactorial etiology. Long-term follow-up data remain limited, underscoring the need for vigilant monitoring. This case reinforces Btx A as a primary therapeutic option and highlights the importance of individualized management in IUTMH. 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引用次数: 0
摘要
孤立性单侧颞肌肥厚(IUTMH)是一种罕见的临床疾病,全球报道的病例不到20例。本病例报告和文献综述旨在扩大对其诊断、管理和结果的理解。一位44岁的阿拉伯亚洲女性,在咀嚼时出现进行性左颞肿胀和疼痛12个月的病史。临床检查发现一个坚硬、无压痛的肿块。影像学[计算机断层扫描(CT)和超声检查]证实孤立性左颞肌肥大,无肿瘤或炎症特征。A型肉毒毒素(Btx A, 25u)注射后症状改善,随访3个月肌肉部分萎缩。对16例已发表病例(包括我们的病例)的文献回顾显示,女性轻微占优势(10/16),不同的症状特征(无痛性肿胀到咀嚼功能障碍)和不同的管理策略。在6/16的病例中,微创Btx A获得了良好的结果,低剂量(25 U)的疗效与高剂量相当。手术干预虽然有效,但有1例复发。诊断依靠影像学(CT/磁共振成像)和组织学(检查时)证实肥厚无恶性。虽然磨牙和压力与一些病例有关,但7/16缺乏可识别的诱因,表明病因是多因素的。长期随访数据仍然有限,这突出了警惕监测的必要性。本病例强调了Btx A作为主要治疗选择的重要性,并强调了IUTMH个体化治疗的重要性。需要进一步的研究来阐明发病机制和优化治疗方案。
Isolated Unilateral Temporalis Muscle Hypertrophy: Comprehensive Literature Review.
Isolated unilateral temporalis muscle hypertrophy (IUTMH) is a rare clinical entity, with fewer than 20 cases reported globally. This case report and literature review aimed to expand the understanding of its diagnosis, management, and outcomes. A 44-year-old Arab Asian woman presented with a 12 month history of progressive left temporal swelling and pain during mastication. Clinical examination revealed a firm, non-tender mass. Imaging [computed tomography (CT) and ultrasonography] confirmed isolated left temporalis muscle hypertrophy without neoplastic or inflammatory features. Botulinum toxin type A (Btx A, 25 U) injection led to symptom improvement and partial muscle reduction at 3 month follow-up. A literature review of 16 published cases (including ours) demonstrated a slight female predominance (10/16), variable symptom profiles (painless swelling to masticatory dysfunction), and diverse management strategies. Minimally-invasive Btx A achieved favorable outcomes in 6/16 cases, with lower doses (25 U) showing efficacy comparable with higher doses. Surgical intervention, although effective, was associated with recurrence in 1 case. Diagnostic reliance on imaging (CT/magnetic resonance imaging) and histology (when performed) confirmed hypertrophy without malignancy. While bruxism and stress were implicated in some cases, 7/16 lacked identifiable triggers, suggesting multifactorial etiology. Long-term follow-up data remain limited, underscoring the need for vigilant monitoring. This case reinforces Btx A as a primary therapeutic option and highlights the importance of individualized management in IUTMH. Further research is warranted to elucidate pathogenesis and optimize treatment protocols.