{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,9]],"date-time":"2026-06-09T15:45:42Z","timestamp":1781019942246,"version":"3.54.1"},"reference-count":24,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2015,7,6]],"date-time":"2015-07-06T00:00:00Z","timestamp":1436140800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"Grant-in-Aid for General Scientific Research from the Ministry of Education, Culture, Sports, Science and Technology, Japan","award":["22590965"],"award-info":[{"award-number":["22590965"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Neuropathology"],"published-print":{"date-parts":[[2015,12]]},"abstract":"<jats:p>Flail arm (FA) syndrome, a minor subtype of amyotrophic lateral sclerosis (ALS), is characterized by progressive weakness and upper girdle wasting, but the associated pathological changes remain unclear. A 59\u2010year\u2010old man was admitted to our hospital with a 3\u2010year history of upper girdle weakness. Bulbar symptom and gait disturbance gradually developed, and he was clinically diagnosed with FA syndrome. After a 10\u2010year disease course, he died of pulmonary adenocarcinoma. Neuropathological examination revealed severe motor neuronal loss in the brain stem and anterior horn of the cervical spinal cord with bilateral pyramidal tract degeneration. The histological findings were consistent with typical ALS, including Bunina bodies and Lewy body\u2010like and skein\u2010like inclusions. Cytoplasmic vacuoles were found in the remaining anterior horn motor neurons of the lumbar spinal cord. This is a unique autopsy case with a long\u2010standing clinical course that suggests that FA syndrome is an atypical form of ALS.<\/jats:p>","DOI":"10.1111\/neup.12223","type":"journal-article","created":{"date-parts":[[2015,7,7]],"date-time":"2015-07-07T02:44:56Z","timestamp":1436237096000},"page":"582-586","source":"Crossref","is-referenced-by-count":6,"title":["Flail arm syndrome with cytoplasmic vacuoles in remaining anterior horn motor neurons: A peculiar variant of amyotrophic lateral sclerosis"],"prefix":"10.1111","volume":"35","author":[{"given":"Shuji","family":"Hino","sequence":"first","affiliation":[{"name":"Department of Neurology Tokyo Women's Medical University  Tokyo Japan"},{"name":"Department of Neurology Saitama Red Cross Hospital  Saitama\u2010City Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shoichi","family":"Sasaki","sequence":"additional","affiliation":[{"name":"Department of Neurology Tokyo Women's Medical University  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2015,7,6]]},"reference":[{"key":"e_1_2_5_2_1","first-page":"1027","article-title":"Amyotrophic lateral sclerosis","volume":"138","author":"Bonduelle M","year":"1982","journal-title":"Rev Neurol (Paris)"},{"key":"e_1_2_5_3_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000049472.47709.3B"},{"key":"e_1_2_5_4_1","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1136\/jnnp.2006.104828","article-title":"Descriptive epidemiology of amyotrophic lateral sclerosis: new evidence and unsolved issues","volume":"79","author":"Logroscino G","year":"2008","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"e_1_2_5_5_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.wnl.0000302187.20239.93"},{"key":"e_1_2_5_6_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.wnl.0000345041.83406.a2"},{"key":"e_1_2_5_7_1","first-page":"346","volume-title":"Maladies du syst\u00e8me nerveux","author":"Vulpian A","year":"1886"},{"key":"e_1_2_5_8_1","first-page":"2355","article-title":"Brachial amyotrophic diplegia: a slowly progressive motor neuron disorder","volume":"54","author":"Gamez J","year":"2000","journal-title":"Neurology"},{"key":"e_1_2_5_9_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.67.2.258"},{"key":"e_1_2_5_10_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.65.6.950"},{"key":"e_1_2_5_11_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.53.5.1071"},{"key":"e_1_2_5_12_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.2010.235952"},{"key":"e_1_2_5_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1468-1331.2004.00841.x"},{"key":"e_1_2_5_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1468-1331.2010.03146.x"},{"key":"e_1_2_5_15_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.66.5.581"},{"key":"e_1_2_5_16_1","doi-asserted-by":"publisher","DOI":"10.1007\/s004010051131"},{"key":"e_1_2_5_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2990.1996.tb00907.x"},{"key":"e_1_2_5_18_1","first-page":"181","article-title":"The comparative neuropathology of motor neuron diseases","volume":"19","author":"Andrews JM","year":"1976","journal-title":"UCLA Forum Med Sci"},{"key":"e_1_2_5_19_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/113.2.497"},{"key":"e_1_2_5_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/0006-8993(95)00063-V"},{"key":"e_1_2_5_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/0896-6273(95)90259-7"},{"key":"e_1_2_5_22_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.18-09-03241.1998"},{"key":"e_1_2_5_23_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.51.3.871"},{"key":"e_1_2_5_24_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.34.4.427"},{"key":"e_1_2_5_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s004010050535"}],"container-title":["Neuropathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fneup.12223","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/neup.12223","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,1]],"date-time":"2023-10-01T22:17:44Z","timestamp":1696198664000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/neup.12223"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,7,6]]},"references-count":24,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2015,12]]}},"alternative-id":["10.1111\/neup.12223"],"URL":"https:\/\/doi.org\/10.1111\/neup.12223","archive":["Portico"],"relation":{},"ISSN":["0919-6544","1440-1789"],"issn-type":[{"value":"0919-6544","type":"print"},{"value":"1440-1789","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,7,6]]}}}