PDF(Pubmed)
Abstract：
BACKGROUND: Isaacs\' syndrome, also known as neuromyotonia or peripheral nerve hyperexcitability, is a rare disorder that affects the peripheral nervous system. Clinical findings include cramps, fasciculations, and myokymia; however, there are few reports of dental treatment for trismus.
METHODS: A patient with trismus due to Isaacs\' syndrome experienced swelling and pain in the gingiva surrounding his right lower first molar. He was diagnosed with chronic apical periodontitis by a dentist near his home. However, the patient was informed that dental treatment and medication could not be administered because of the presence of Isaacs\' syndrome, and he visited the Geriatric Dentistry and Perioperative Oral Care Center at Kyushu University Hospital 2 weeks later. The patient\'s painless mouth-opening distance (between incisors) was 20 mm at that time, and medication, including amoxicillin capsules and acetaminophen, was administered because the dental extraction forceps or endodontic instruments were difficult to insert into the oral cavity for treatment. Two months after his initial visit, the patient visited us complaining of pain in the same area. However, he had recently undergone plasmapheresis treatment in neurology to alleviate limited mouth opening and systemic myalgia, resulting in a pain-free mouth-opening distance of approximately 35 mm. During this temporary period in which he had no restriction in mouth opening, we performed tooth extraction and bridge restoration on the mandibular right first molar and created an oral appliance for sleep bruxism.
CONCLUSIONS: Plasmapheresis therapy transiently reduced trismus, rendering dental interventions feasible, albeit temporarily. This case report underscores the importance of close collaboration between neurologists and dentists who encounter similar cases while furnishing valuable insights to inform dental treatment planning.
METHODS: A patient with trismus due to Isaacs\' syndrome experienced swelling and pain in the gingiva surrounding his right lower first molar. He was diagnosed with chronic apical periodontitis by a dentist near his home. However, the patient was informed that dental treatment and medication could not be administered because of the presence of Isaacs\' syndrome, and he visited the Geriatric Dentistry and Perioperative Oral Care Center at Kyushu University Hospital 2 weeks later. The patient\'s painless mouth-opening distance (between incisors) was 20 mm at that time, and medication, including amoxicillin capsules and acetaminophen, was administered because the dental extraction forceps or endodontic instruments were difficult to insert into the oral cavity for treatment. Two months after his initial visit, the patient visited us complaining of pain in the same area. However, he had recently undergone plasmapheresis treatment in neurology to alleviate limited mouth opening and systemic myalgia, resulting in a pain-free mouth-opening distance of approximately 35 mm. During this temporary period in which he had no restriction in mouth opening, we performed tooth extraction and bridge restoration on the mandibular right first molar and created an oral appliance for sleep bruxism.
CONCLUSIONS: Plasmapheresis therapy transiently reduced trismus, rendering dental interventions feasible, albeit temporarily. This case report underscores the importance of close collaboration between neurologists and dentists who encounter similar cases while furnishing valuable insights to inform dental treatment planning.
摘要：
背景：艾萨克综合征，也称为神经肌强直或周围神经过度兴奋，是一种影响周围神经系统的罕见疾病。临床发现包括抽筋,束感，和Myokymia；然而，很少有关于牙关治疗的报道。
方法：一名因Isaacs\'综合征而出现了三联肌的患者，其右下第一磨牙周围的牙龈出现肿胀和疼痛。他在家附近的一名牙医诊断出患有慢性根尖周炎。然而,患者被告知，由于存在艾萨克综合征，无法进行牙科治疗和药物治疗，两周后，他访问了九州大学医院的老年牙科和围手术期口腔护理中心。当时患者的无痛张口距离（门牙之间）为20毫米，和药物,包括阿莫西林胶囊和对乙酰氨基酚,因为拔牙钳或牙髓器械很难插入口腔进行治疗。在他初次访问两个月后，病人来看望我们，抱怨同一区域疼痛。然而,他最近在神经科接受了血浆置换治疗，以缓解张口受限和全身肌痛，导致无痛的张口距离约为35毫米。在这个临时时期，他没有张嘴的限制，我们在下颌右第一磨牙上进行了拔牙和牙桥修复，并创建了一种用于睡眠磨牙症的口腔矫治器。
结论：血浆置换疗法可短暂减少三联肌，使牙科干预可行，虽然是暂时的。本病例报告强调了神经学家和遇到类似病例的牙医之间密切合作的重要性，同时提供有价值的见解以告知牙科治疗计划。
方法：一名因Isaacs\'综合征而出现了三联肌的患者，其右下第一磨牙周围的牙龈出现肿胀和疼痛。他在家附近的一名牙医诊断出患有慢性根尖周炎。然而,患者被告知，由于存在艾萨克综合征，无法进行牙科治疗和药物治疗，两周后，他访问了九州大学医院的老年牙科和围手术期口腔护理中心。当时患者的无痛张口距离（门牙之间）为20毫米，和药物,包括阿莫西林胶囊和对乙酰氨基酚,因为拔牙钳或牙髓器械很难插入口腔进行治疗。在他初次访问两个月后，病人来看望我们，抱怨同一区域疼痛。然而,他最近在神经科接受了血浆置换治疗，以缓解张口受限和全身肌痛，导致无痛的张口距离约为35毫米。在这个临时时期，他没有张嘴的限制，我们在下颌右第一磨牙上进行了拔牙和牙桥修复，并创建了一种用于睡眠磨牙症的口腔矫治器。
结论：血浆置换疗法可短暂减少三联肌，使牙科干预可行，虽然是暂时的。本病例报告强调了神经学家和遇到类似病例的牙医之间密切合作的重要性，同时提供有价值的见解以告知牙科治疗计划。
