{"id":"https://openalex.org/W7223317779","doi":"https://doi.org/10.3390/jcm15218217","title":"Anesthetic Management of Stiff-Person Syndrome During Prolonged Lateral Skull Base Surgery: A Case Report and Phenotype-Informed Review","display_name":"Anesthetic Management of Stiff-Person Syndrome During Prolonged Lateral Skull Base Surgery: A Case Report and Phenotype-Informed Review","publication_year":2026,"publication_date":"2026-10-22","ids":{"openalex":"https://openalex.org/W7223317779","doi":"https://doi.org/10.3390/jcm15218217"},"language":"en","primary_location":{"id":"doi:10.3390/jcm15218217","doi":"https://doi.org/10.3390/jcm15218217","is_oa":true,"landing_page_url":"https://doi.org/10.3390/jcm15218217","pdf_url":null,"source":{"id":"https://openalex.org/S2737969411","display_name":"Journal of Clinical Medicine","issn_l":"2077-0383","issn":["2077-0383"],"is_oa":true,"is_in_doaj":false,"is_core":true,"listed_in":["cwts-core","ki-jl-1","norway-1","poland-140","russia-white-list-1","vabb-shw"],"host_organization":"https://openalex.org/P4310310987","host_organization_name":"Multidisciplinary Digital Publishing Institute","host_organization_lineage":["https://openalex.org/P4310310987"],"host_organization_lineage_names":["Multidisciplinary Digital Publishing Institute"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Clinical Medicine","raw_type":"journal-article"},"volume":null,"type":"article","is_traditional":true,"indexed_in":["crossref"],"open_access":{"is_oa":true,"oa_status":"gold","oa_url":"https://doi.org/10.3390/jcm15218217","any_repository_has_fulltext":false},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5158787349","display_name":"Jianhao Zhang","orcid":"https://orcid.org/0009-0009-8198-5625","observed_orcids":["https://orcid.org/0009-0009-8198-5625"]},"institutions":[{"id":"https://openalex.org/I200296433","display_name":"Chinese Academy of Medical Sciences & Peking Union Medical College","ror":"https://ror.org/02drdmm93","country_code":"CN","type":"education","lineage":["https://openalex.org/I200296433"]},{"id":"https://openalex.org/I2801228662","display_name":"Peking Union Medical College Hospital","ror":"https://ror.org/04jztag35","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I2801228662"]},{"id":"https://openalex.org/I4210092004","display_name":"Plastic Surgery Hospital","ror":"https://ror.org/00fk0yb75","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I4210092004"]}],"countries":["CN"],"is_corresponding":false,"raw_author_name":"Jianhao Zhang","raw_affiliation_strings":["Department of Anesthesiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China","Department of Anesthesiology, Plastic Surgery Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100144, China"],"raw_orcid":"https://orcid.org/0009-0009-8198-5625","affiliations":[{"raw_affiliation_string":"Department of Anesthesiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China","institution_ids":["https://openalex.org/I200296433","https://openalex.org/I2801228662"]},{"raw_affiliation_string":"Department of Anesthesiology, Plastic Surgery Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100144, China","institution_ids":["https://openalex.org/I200296433","https://openalex.org/I4210092004"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5136688311","display_name":"Haisong Lu","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I200296433","display_name":"Chinese Academy of Medical Sciences & Peking Union Medical College","ror":"https://ror.org/02drdmm93","country_code":"CN","type":"education","lineage":["https://openalex.org/I200296433"]},{"id":"https://openalex.org/I2801228662","display_name":"Peking Union Medical College Hospital","ror":"https://ror.org/04jztag35","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I2801228662"]}],"countries":["CN"],"is_corresponding":false,"raw_author_name":"Haisong Lu","raw_affiliation_strings":["Department of Anesthesiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Anesthesiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China","institution_ids":["https://openalex.org/I200296433","https://openalex.org/I2801228662"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5103935727","display_name":"Zhao Yang","orcid":"https://orcid.org/0000-0002-5880-9465","observed_orcids":["https://orcid.org/0000-0002-5880-9465"]},"institutions":[{"id":"https://openalex.org/I200296433","display_name":"Chinese Academy of Medical Sciences & Peking Union Medical College","ror":"https://ror.org/02drdmm93","country_code":"CN","type":"education","lineage":["https://openalex.org/I200296433"]},{"id":"https://openalex.org/I2801228662","display_name":"Peking Union Medical College Hospital","ror":"https://ror.org/04jztag35","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I2801228662"]}],"countries":["CN"],"is_corresponding":false,"raw_author_name":"Yang Zhao","raw_affiliation_strings":["Department of Otolaryngology\u2013Head and Neck Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China"],"raw_orcid":"https://orcid.org/0000-0002-5880-9465","affiliations":[{"raw_affiliation_string":"Department of Otolaryngology\u2013Head and Neck Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China","institution_ids":["https://openalex.org/I200296433","https://openalex.org/I2801228662"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5101696073","display_name":"Guodong Feng","orcid":"https://orcid.org/0000-0002-2292-2466","observed_orcids":["https://orcid.org/0000-0002-2292-2466"]},"institutions":[{"id":"https://openalex.org/I200296433","display_name":"Chinese Academy of Medical Sciences & Peking Union Medical College","ror":"https://ror.org/02drdmm93","country_code":"CN","type":"education","lineage":["https://openalex.org/I200296433"]},{"id":"https://openalex.org/I2801228662","display_name":"Peking Union Medical College Hospital","ror":"https://ror.org/04jztag35","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I2801228662"]}],"countries":["CN"],"is_corresponding":false,"raw_author_name":"Guodong Feng","raw_affiliation_strings":["Department of Otolaryngology\u2013Head and Neck Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Otolaryngology\u2013Head and Neck Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China","institution_ids":["https://openalex.org/I200296433","https://openalex.org/I2801228662"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5007593609","display_name":"Yuguan Zhang","orcid":"https://orcid.org/0000-0003-4149-8370","observed_orcids":["https://orcid.org/0000-0003-4149-8370"]},"institutions":[{"id":"https://openalex.org/I200296433","display_name":"Chinese Academy of Medical Sciences & Peking Union Medical College","ror":"https://ror.org/02drdmm93","country_code":"CN","type":"education","lineage":["https://openalex.org/I200296433"]},{"id":"https://openalex.org/I2801228662","display_name":"Peking Union Medical College Hospital","ror":"https://ror.org/04jztag35","country_code":"CN","type":"healthcare","lineage":["https://openalex.org/I2801228662"]}],"countries":["CN"],"is_corresponding":true,"raw_author_name":"Yuguan Zhang","raw_affiliation_strings":["Department of Anesthesiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China"],"raw_orcid":"https://orcid.org/0000-0003-4149-8370","affiliations":[{"raw_affiliation_string":"Department of Anesthesiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China","institution_ids":["https://openalex.org/I200296433","https://openalex.org/I2801228662"]}]}],"institutions":[],"countries_distinct_count":1,"institutions_distinct_count":3,"corresponding_author_ids":["https://openalex.org/A5007593609"],"corresponding_institution_ids":["https://openalex.org/I200296433","https://openalex.org/I2801228662"],"apc_list":{"value":2600,"currency":"CHF","value_usd":3137},"apc_paid":{"value":2600,"currency":"CHF","value_usd":3137},"fwci":0.0,"has_fulltext":false,"cited_by_count":0,"citation_normalized_percentile":{"value":0.85968871,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":null,"biblio":{"volume":"15","issue":"21","first_page":"8217","last_page":"8217"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T10436","display_name":"Anesthesia and Sedative Agents","score":0.9291999936103821,"subfield":{"id":"https://openalex.org/subfields/2703","display_name":"Anesthesiology and Pain Medicine"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},"topics":[{"id":"https://openalex.org/T10436","display_name":"Anesthesia and Sedative Agents","score":0.9291999936103821,"subfield":{"id":"https://openalex.org/subfields/2703","display_name":"Anesthesiology and Pain Medicine"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T10189","display_name":"Anesthesia and Pain Management","score":0.029200000688433647,"subfield":{"id":"https://openalex.org/subfields/2703","display_name":"Anesthesiology and Pain Medicine"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T11627","display_name":"Autoimmune Neurological Disorders and Treatments","score":0.011599999852478504,"subfield":{"id":"https://openalex.org/subfields/2728","display_name":"Clinical Neurology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/stiff-person-syndrome","display_name":"stiff person syndrome","score":0.9980000257492065},{"id":"https://openalex.org/keywords/lateral-skull-base-surgery","display_name":"lateral skull base surgery","score":0.906000018119812},{"id":"https://openalex.org/keywords/total-intravenous-anesthesia","display_name":"total intravenous anesthesia","score":0.925000011920929},{"id":"https://openalex.org/keywords/muscle-relaxant-free-anesthesia","display_name":"muscle relaxant-free anesthesia","score":0.8019999861717224},{"id":"https://openalex.org/keywords/anti-amphiphysin-antibodies","display_name":"anti-amphiphysin antibodies","score":0.8040000200271606},{"id":"https://openalex.org/keywords/quantitative-neuromuscular-monitoring","display_name":"quantitative neuromuscular monitoring","score":0.7749999761581421},{"id":"https://openalex.org/keywords/train-of-four-monitoring","display_name":"train-of-four monitoring","score":0.7960000038146973},{"id":"https://openalex.org/keywords/bispectral-index","display_name":"bispectral index","score":0.8640000224113464},{"id":"https://openalex.org/keywords/perioperative-planning","display_name":"perioperative planning","score":0.7099999785423279},{"id":"https://openalex.org/keywords/phosphaturic-mesenchymal-tumor","display_name":"phosphaturic mesenchymal tumor","score":0.7580000162124634},{"id":"https://openalex.org/keywords/autoimmune-neurological-disorders","display_name":"autoimmune neurological disorders","score":0.38199999928474426}],"concepts":[{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.9761999845504761},{"id":"https://openalex.org/C42219234","wikidata":"https://www.wikidata.org/wiki/Q131130","display_name":"Anesthesia","level":1,"score":0.7240999937057495},{"id":"https://openalex.org/C31174226","wikidata":"https://www.wikidata.org/wiki/Q64855140","display_name":"Perioperative","level":2,"score":0.7128000259399414},{"id":"https://openalex.org/C2776376669","wikidata":"https://www.wikidata.org/wiki/Q5094647","display_name":"Intensive care unit","level":2,"score":0.548799991607666},{"id":"https://openalex.org/C2779300802","wikidata":"https://www.wikidata.org/wiki/Q13147","display_name":"Skull","level":2,"score":0.5333999991416931},{"id":"https://openalex.org/C2778162923","wikidata":"https://www.wikidata.org/wiki/Q4990531","display_name":"Anesthetic","level":2,"score":0.5045999884605408},{"id":"https://openalex.org/C141071460","wikidata":"https://www.wikidata.org/wiki/Q40821","display_name":"Surgery","level":1,"score":0.49549999833106995},{"id":"https://openalex.org/C2781097354","wikidata":"https://www.wikidata.org/wiki/Q4918396","display_name":"Bispectral index","level":3,"score":0.4932999908924103},{"id":"https://openalex.org/C12770488","wikidata":"https://www.wikidata.org/wiki/Q188449","display_name":"Neurosurgery","level":2,"score":0.38670000433921814},{"id":"https://openalex.org/C2779526319","wikidata":"https://www.wikidata.org/wiki/Q615057","display_name":"Anesthesiology","level":2,"score":0.3813999891281128},{"id":"https://openalex.org/C2779358675","wikidata":"https://www.wikidata.org/wiki/Q3766250","display_name":"Respiratory monitoring","level":3,"score":0.3774999976158142},{"id":"https://openalex.org/C2781302539","wikidata":"https://www.wikidata.org/wiki/Q412133","display_name":"Dexmedetomidine","level":3,"score":0.3589000105857849},{"id":"https://openalex.org/C16568411","wikidata":"https://www.wikidata.org/wiki/Q83042","display_name":"Neurology","level":2,"score":0.3285999894142151},{"id":"https://openalex.org/C2778165595","wikidata":"https://www.wikidata.org/wiki/Q5491454","display_name":"Resuscitation","level":2,"score":0.3109999895095825},{"id":"https://openalex.org/C2777449187","wikidata":"https://www.wikidata.org/wiki/Q575168","display_name":"Hyperventilation","level":2,"score":0.3061000108718872},{"id":"https://openalex.org/C2780954326","wikidata":"https://www.wikidata.org/wiki/Q1416096","display_name":"Intracranial pressure","level":2,"score":0.288100004196167},{"id":"https://openalex.org/C108516343","wikidata":"https://www.wikidata.org/wiki/Q189553","display_name":"Otorhinolaryngology","level":2,"score":0.263700008392334},{"id":"https://openalex.org/C178853913","wikidata":"https://www.wikidata.org/wiki/Q1642137","display_name":"Hemodynamics","level":2,"score":0.25780001282691956},{"id":"https://openalex.org/C2909465803","wikidata":"https://www.wikidata.org/wiki/Q3286619","display_name":"Neuromuscular Blockade","level":2,"score":0.2574999928474426},{"id":"https://openalex.org/C2780795376","wikidata":"https://www.wikidata.org/wiki/Q243547","display_name":"Ketamine","level":2,"score":0.2533000111579895}],"mesh":[],"locations_count":1,"locations":[{"id":"doi:10.3390/jcm15218217","doi":"https://doi.org/10.3390/jcm15218217","is_oa":true,"landing_page_url":"https://doi.org/10.3390/jcm15218217","pdf_url":null,"source":{"id":"https://openalex.org/S2737969411","display_name":"Journal of Clinical Medicine","issn_l":"2077-0383","issn":["2077-0383"],"is_oa":true,"is_in_doaj":false,"is_core":true,"listed_in":["cwts-core","ki-jl-1","norway-1","poland-140","russia-white-list-1","vabb-shw"],"host_organization":"https://openalex.org/P4310310987","host_organization_name":"Multidisciplinary Digital Publishing Institute","host_organization_lineage":["https://openalex.org/P4310310987"],"host_organization_lineage_names":["Multidisciplinary Digital Publishing Institute"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Clinical Medicine","raw_type":"journal-article"}],"best_oa_location":{"id":"doi:10.3390/jcm15218217","doi":"https://doi.org/10.3390/jcm15218217","is_oa":true,"landing_page_url":"https://doi.org/10.3390/jcm15218217","pdf_url":null,"source":{"id":"https://openalex.org/S2737969411","display_name":"Journal of Clinical Medicine","issn_l":"2077-0383","issn":["2077-0383"],"is_oa":true,"is_in_doaj":false,"is_core":true,"listed_in":["cwts-core","ki-jl-1","norway-1","poland-140","russia-white-list-1","vabb-shw"],"host_organization":"https://openalex.org/P4310310987","host_organization_name":"Multidisciplinary Digital Publishing Institute","host_organization_lineage":["https://openalex.org/P4310310987"],"host_organization_lineage_names":["Multidisciplinary Digital Publishing Institute"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Clinical Medicine","raw_type":"journal-article"},"sustainable_development_goals":[{"display_name":"Good health and well-being","id":"https://metadata.un.org/sdg/3","score":0.9940000176429749}],"study_designs":[{"display_name":"Case Report","id":"https://openalex.org/study-designs/case-report"}],"awards":[],"funders":[{"id":"https://openalex.org/F4320332531","display_name":"Wu Jieping Medical Foundation","ror":"https://ror.org/05dgwj702"}],"has_content":{"grobid_xml":false,"pdf":false},"content_urls":null,"referenced_works_count":24,"referenced_works":["https://openalex.org/W126727042","https://openalex.org/W1966718339","https://openalex.org/W1973487832","https://openalex.org/W1991105627","https://openalex.org/W2071458642","https://openalex.org/W2110542170","https://openalex.org/W2140308853","https://openalex.org/W2316040471","https://openalex.org/W2333301957","https://openalex.org/W2339045727","https://openalex.org/W2341543030","https://openalex.org/W2417346536","https://openalex.org/W2799492095","https://openalex.org/W2977415948","https://openalex.org/W2979555467","https://openalex.org/W3163926470","https://openalex.org/W4254473047","https://openalex.org/W4292358512","https://openalex.org/W4297925893","https://openalex.org/W4311279646","https://openalex.org/W4377221479","https://openalex.org/W4391923636","https://openalex.org/W4402169024","https://openalex.org/W7203479731"],"related_works":["https://openalex.org/W2408900424","https://openalex.org/W4413416614","https://openalex.org/W1990639492","https://openalex.org/W126727042","https://openalex.org/W2129824926","https://openalex.org/W4292358512","https://openalex.org/W4403149853","https://openalex.org/W3032131582","https://openalex.org/W137882974","https://openalex.org/W4414581474"],"abstract_inverted_index":{"Background:":[0],"Stiff-person":[1],"syndrome":[2],"(SPS)":[3],"is":[4,43],"a":[5,72,221],"rare":[6],"autoimmune":[7],"neurological":[8],"disorder":[9,34],"characterized":[10],"by":[11],"progressive":[12],"rigidity":[13],"and":[14,71,90,100,114,148,170,205,219],"episodic":[15],"spasms.":[16],"Perioperative":[17,190],"evidence":[18],"consists":[19],"mainly":[20],"of":[21,27,48,124,158],"case":[22],"reports,":[23],"underscoring":[24],"the":[25,31,44,127],"importance":[26],"incorporating":[28],"heterogeneity":[29],"across":[30],"stiff-person":[32],"spectrum":[33],"(SPSD)":[35],"into":[36],"perioperative":[37,217],"assessment.":[38],"To":[39],"our":[40],"knowledge,":[41],"this":[42,186],"first":[45],"published":[46,159],"report":[47],"SPS":[49,169,188],"management":[50],"during":[51],"prolonged":[52,172],"lateral":[53,164],"skull":[54,73,165],"base":[55,74,166],"surgery.":[56],"Case":[57],"Presentation:":[58],"A":[59,155],"56-year-old":[60],"man":[61],"with":[62,87,122],"anti-amphiphysin":[63],"antibody-positive,":[64],"lower-limb-predominant":[65],"SPSD":[66,206],"without":[67,94],"documented":[68],"generalized":[69],"spasms":[70],"phosphaturic":[75],"mesenchymal":[76],"tumor":[77],"underwent":[78],"an":[79],"11":[80],"h":[81,142],"radical":[82],"resection.":[83],"Total":[84],"intravenous":[85,181],"anesthesia":[86,182],"propofol,":[88],"remifentanil,":[89],"dexmedetomidine":[91],"was":[92,105,139,183],"used":[93],"neuromuscular":[95,174],"blocking":[96],"agents":[97],"because":[98],"facial":[99],"lower":[101],"cranial":[102],"nerve":[103],"monitoring":[104,118,175],"required.":[106],"Bispectral":[107],"index":[108],"values":[109],"were":[110],"maintained":[111],"at":[112],"30\u201350,":[113],"serial":[115],"quantitative":[116,173],"train-of-four":[117],"showed":[119],"four":[120],"responses":[121],"ratios":[123],"100\u2013125%":[125],"throughout":[126],"procedure.":[128],"No":[129],"intraoperative":[130],"spasm,":[131],"movement,":[132],"or":[133,198],"hemodynamic":[134],"instability":[135],"occurred.":[136],"The":[137,208],"patient":[138],"extubated":[140],"3.5":[141],"after":[143],"intensive":[144],"care":[145],"unit":[146],"admission,":[147],"serum":[149],"phosphorus":[150],"normalized":[151],"within":[152],"48":[153],"h.":[154],"focused":[156],"review":[157],"reports":[160],"identified":[161],"no":[162],"previous":[163],"surgery":[167],"in":[168,185],"limited":[171],"data.":[176],"Conclusions:":[177],"Prolonged":[178],"muscle-relaxant-free":[179],"total":[180],"feasible":[184],"selected":[187],"phenotype.":[189,207],"planning":[191,218],"should":[192],"integrate":[193],"individual":[194],"spasm":[195],"history,":[196],"bulbar":[197],"respiratory":[199],"involvement,":[200],"medication":[201],"burden,":[202],"surgical":[203],"requirements,":[204],"proposed":[209],"phenotype-informed":[210],"framework":[211],"organizes":[212],"these":[213],"considerations":[214],"for":[215,223],"individualized":[216],"provides":[220],"basis":[222],"future":[224],"evaluation.":[225]},"counts_by_year":[],"updated_date":"2026-11-04T10:54:05.112534","created_date":"2026-10-24T00:00:00"}
