{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2024,11,23]],"date-time":"2024-11-23T05:29:23Z","timestamp":1732339763236,"version":"3.28.0"},"reference-count":16,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2024,6,22]],"date-time":"2024-06-22T00:00:00Z","timestamp":1719014400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,6,22]],"date-time":"2024-06-22T00:00:00Z","timestamp":1719014400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Oral Health"],"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Isaacs\u2019 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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation<\/jats:title><jats:p>A patient with trismus due to Isaacs\u2019 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\u2019 syndrome, and he visited the Geriatric Dentistry and Perioperative Oral Care Center at Kyushu University Hospital 2 weeks later. The patient\u2019s painless mouth-opening distance (between incisors) was 20\u00a0mm 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\u00a0mm. 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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>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.<\/jats:p><\/jats:sec>","DOI":"10.1186\/s12903-024-04485-2","type":"journal-article","created":{"date-parts":[[2024,6,22]],"date-time":"2024-06-22T06:02:16Z","timestamp":1719036136000},"update-policy":"http:\/\/dx.doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Oral management for a patient with trismus accompanied by Isaacs\u2019 syndrome: a case report"],"prefix":"10.1186","volume":"24","author":[{"given":"Asuka","family":"Tani","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shinsuke","family":"Mizutani","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mitsuru","family":"Watanabe","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takashi","family":"Irie","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Katsuhisa","family":"Masaki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Noriko","family":"Isobe","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Haruhiko","family":"Kashiwazaki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2024,6,22]]},"reference":[{"key":"4485_CR1","doi-asserted-by":"publisher","first-page":"1830","DOI":"10.1016\/j.clinph.2021.03.053","volume":"132","author":"J Bashford","year":"2021","unstructured":"Bashford J, Chan WK, Coutinho E, Norwood F, Mills K, Shaw CE. Demystifying the spontaneous phenomena of motor hyperexcitability. Clin Neurophysiol. 2021;132:1830\u201344.","journal-title":"Clin Neurophysiol"},{"key":"4485_CR2","doi-asserted-by":"publisher","first-page":"5","DOI":"10.1002\/mus.24632","volume":"52","author":"A Ahmed","year":"2015","unstructured":"Ahmed A, Simmons Z. Isaacs syndrome: a review. Muscle Nerve. 2015;52:5\u201312.","journal-title":"Muscle Nerve"},{"key":"4485_CR3","unstructured":"Orphanet. Issacs syndrome. 2013. https:\/\/www.orpha.net\/consor\/cgi-bin\/OC_Exp.php?lng=EN&Expert=84142. Accessed 28 Dec 2023."},{"key":"4485_CR4","doi-asserted-by":"crossref","first-page":"117","DOI":"10.4103\/ajts.ajts_88_21","volume":"17","author":"PP Tripathi","year":"2023","unstructured":"Tripathi PP, Kumari S, Prabhat N, Lamba DS, Hans R, Goyal MK, et al. Effectiveness of therapeutic plasma exchange in case of rare neurological disorder Isaacs syndrome. Asian J Transfus Sci. 2023;17:117\u201320.","journal-title":"Asian J Transfus Sci"},{"key":"4485_CR5","doi-asserted-by":"publisher","first-page":"267","DOI":"10.1002\/jca.21233","volume":"27","author":"EA Jaben","year":"2012","unstructured":"Jaben EA, Winters JL. Plasma exchange as a therapeutic option in patients with neurologic symptoms due to antibodies to voltage-gated potassium channels: a report of five cases and review of the literature. J Clin Apher. 2012;27:267\u201373.","journal-title":"J Clin Apher"},{"key":"4485_CR6","doi-asserted-by":"publisher","first-page":"74","DOI":"10.1186\/s12883-022-02584-7","volume":"22","author":"KC Li","year":"2022","unstructured":"Li KC, Liao MF, Wu YR, Lyu RK. Isaacs\u2019 syndrome as the initial presentation of malignant thymoma and associated with double-positive voltage-gated potassium channel complex antibodies, a case report. BMC Neurol. 2022;22:74.","journal-title":"BMC Neurol"},{"key":"4485_CR7","first-page":"e30100","volume":"14","author":"K Horiuchi","year":"2022","unstructured":"Horiuchi K, Kudo A, Inoue T, Fujii S, Oshima Y. Rituximab was effective in relieving symptoms of Isaacs syndrome: a case report. Cureus. 2022;14:e30100.","journal-title":"Cureus"},{"key":"4485_CR8","doi-asserted-by":"publisher","first-page":"814","DOI":"10.1016\/j.jocn.2009.10.025","volume":"17","author":"VK Paliwal","year":"2010","unstructured":"Paliwal VK. Neuromyotonia masquerading as tetanus. J Clin Neurosci. 2010;17:814\u20135.","journal-title":"J Clin Neurosci"},{"key":"4485_CR9","doi-asserted-by":"publisher","first-page":"56","DOI":"10.1002\/jgf2.302","volume":"21","author":"K Ota","year":"2020","unstructured":"Ota K, Fukui K, Nakamura E, Oka M, Ota K, Sakaue M, et al. Effect of Shakuyaku-Kanzo-to in patients with muscle cramps: a systematic literature review. J Gen Fam Med. 2020;21:56\u201362.","journal-title":"J Gen Fam Med"},{"key":"4485_CR10","doi-asserted-by":"publisher","first-page":"140","DOI":"10.1111\/iej.13835","volume":"56","author":"H Fransson","year":"2023","unstructured":"Fransson H, Dawson V. Tooth survival after endodontic treatment. Int Endod J. 2023;56:140\u201353.","journal-title":"Int Endod J"},{"key":"4485_CR11","doi-asserted-by":"publisher","first-page":"81","DOI":"10.1111\/joor.12558","volume":"45","author":"L Jokubauskas","year":"2018","unstructured":"Jokubauskas L, Baltru\u0161aityt\u0117 A, Pilei\u010dikien\u0117 G. Oral appliances for managing sleep bruxism in adults: a systematic review from 2007 to 2017. J Oral Rehabil. 2018;45:81\u201395.","journal-title":"J Oral Rehabil"},{"key":"4485_CR12","doi-asserted-by":"publisher","first-page":"66","DOI":"10.1159\/000528556","volume":"48","author":"C Altobelli","year":"2023","unstructured":"Altobelli C, Anastasio P, Cerrone A, Signoriello E, Lus G, Pluvio C, et al. Therapeutic plasmapheresis: a revision of literature. Kidney Blood Press Res. 2023;48:66\u201378.","journal-title":"Kidney Blood Press Res"},{"key":"4485_CR13","first-page":"1601","volume":"48","author":"H Isaacs","year":"1974","unstructured":"Isaacs H, Frere G. Syndrome of continuous muscle fibre activity. Histochemical, nerve terminal and end-plate study of two cases. S Afr Med J. 1974;48:1601\u20137.","journal-title":"S Afr Med J"},{"key":"4485_CR14","doi-asserted-by":"publisher","first-page":"398","DOI":"10.1177\/154405910408300509","volume":"83","author":"C Dub\u00e9","year":"2004","unstructured":"Dub\u00e9 C, Rompr\u00e9 PH, Manzini C, Guitard F, de Grandmont P, Lavigne GJ. Quantitative polygraphic controlled study on efficacy and safety of oral splint devices in tooth-grinding subjects. J Dent Res. 2004;83:398\u2013403.","journal-title":"J Dent Res"},{"key":"4485_CR15","doi-asserted-by":"publisher","first-page":"482","DOI":"10.1111\/j.1365-2842.2005.01576.x","volume":"33","author":"T Harada","year":"2006","unstructured":"Harada T, Ichiki R, Tsukiyama Y, Koyano K. The effect of oral splint devices on sleep bruxism: a 6-week observation with an ambulatory electromyographic recording device. J Oral Rehabil. 2006;33:482\u20138.","journal-title":"J Oral Rehabil"},{"key":"4485_CR16","doi-asserted-by":"publisher","first-page":"105","DOI":"10.1186\/s10194-020-01160-z","volume":"21","author":"H Jasim","year":"2020","unstructured":"Jasim H, Ghafouri B, Gerdle B, Hedenberg-Magnusson B, Ernberg M. Altered levels of salivary and plasma pain related markers in temporomandibular disorders. J Headache Pain. 2020;21:105.","journal-title":"J Headache Pain"}],"container-title":["BMC Oral Health"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12903-024-04485-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12903-024-04485-2\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12903-024-04485-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,11,22]],"date-time":"2024-11-22T12:07:56Z","timestamp":1732277276000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcoralhealth.biomedcentral.com\/articles\/10.1186\/s12903-024-04485-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,6,22]]},"references-count":16,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,12]]}},"alternative-id":["4485"],"URL":"https:\/\/doi.org\/10.1186\/s12903-024-04485-2","relation":{},"ISSN":["1472-6831"],"issn-type":[{"type":"electronic","value":"1472-6831"}],"subject":[],"published":{"date-parts":[[2024,6,22]]},"assertion":[{"value":"12 March 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"14 June 2024","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"22 June 2024","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Ethical approval is not applicable but this case report was conducted in accordance with the Declaration of Helsinki regarding medical protocols and ethics. Informed written consent was obtained from the patient before the participation in this study.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Informed written consent was obtained from the patient to publish the medical history, clinical photos, and radiographs, all documented in his medical chart.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"716"}}