{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T05:28:30Z","timestamp":1767677310606,"version":"3.48.0"},"reference-count":27,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T00:00:00Z","timestamp":1767657600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:p>Restless Legs Syndrome (RLS) is a common neurological disorder characterized by an irresistible urge to move the legs, often accompanied by uncomfortable sensations. These symptoms typically worsen during periods of rest or in the evening and are partially or temporarily relieved by movement. Variant RLS is diagnosed when these core symptoms extend to extra-leg regions\u2014such as the arms, abdomen, or bladder\u2014while still fulfilling the diagnostic criteria established by the International Restless Legs Syndrome Study Group. We report a case of an adult female patient with refractory right shoulder pain, ultimately diagnosed as variant RLS. Initially misdiagnosed as frozen shoulder, the correct diagnosis was confirmed through polysomnography, which revealed an elevated Periodic Limb Movement during Sleep index, and a supportive International RLS Rating Scale score. Following targeted treatment involving iron supplements for iron deficiency anemia and pramipexole for symptomatic relief, the patient\u2019s shoulder pain and other associated symptoms improved markedly. This case expands the known clinical spectrum of variant RLS by illustrating a pain-dominant upper limb phenotype. Furthermore, we provide a summary analysis of previously reported cases of restless arms syndrome, highlighting the importance of including variant RLS in the differential diagnosis for patients presenting with unexplained shoulder or arm discomfort.<\/jats:p>","DOI":"10.3389\/fmed.2025.1725747","type":"journal-article","created":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T05:19:35Z","timestamp":1767676775000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Variant restless legs syndrome masquerading as refractory shoulder pain: a case report and literature review"],"prefix":"10.3389","volume":"12","author":[{"given":"Peng","family":"Wang","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ling","family":"Yu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Liang","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jianyu","family":"Ren","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rongwei","family":"Qin","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Guohong","family":"Luo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Siyu","family":"Chen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2026,1,6]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"283","DOI":"10.1016\/j.smrv.2011.05.002","article-title":"Epidemiology of restless legs syndrome: a synthesis of the literature","volume":"16","author":"Ohayon","year":"2012","journal-title":"Sleep Med Rev"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"860","DOI":"10.1016\/j.sleep.2014.03.025","article-title":"Restless legs syndrome\/Willis-ekbom disease diagnostic criteria: updated international restless legs syndrome study group (IRLSSG) consensus criteria--history, rationale, description, and significance","volume":"15","author":"Allen","year":"2014","journal-title":"Sleep Med"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"e28896","DOI":"10.1016\/j.heliyon.2024.e28896","article-title":"Restless legs syndrome variants: a systematic review","volume":"10","author":"Haghshenas","year":"2024","journal-title":"Heliyon"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"822","DOI":"10.1001\/jama.236.7.822c","article-title":"Letter: restless arms syndrome","volume":"236","author":"Webb","year":"1976","journal-title":"JAMA"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"959","DOI":"10.1212\/WNL.0000000000001306","article-title":"Bedtime-related jerks in the upper limbs associated with restless arms syndrome","volume":"84","author":"Ruppert","year":"2015","journal-title":"Neurology"},{"key":"ref6","doi-asserted-by":"publisher","first-page":"80","DOI":"10.1038\/s41572-021-00311-z","article-title":"Restless legs syndrome","volume":"7","author":"Manconi","year":"2021","journal-title":"Nat Rev Dis Primers"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"711","DOI":"10.5692\/clinicalneurol.cn-001048","article-title":"A 70-year-old woman presenting with restless shoulder following posterior internal capsule infarction","volume":"57","author":"Matsubara","year":"2017","journal-title":"Rinsho Shinkeigaku"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"317","DOI":"10.1002\/mdc3.12088","article-title":"Review of severity rating scales for restless legs syndrome: critique and recommendations","volume":"1","author":"Walters","year":"2014","journal-title":"Mov Disord Clin Pract"},{"key":"ref9","doi-asserted-by":"publisher","first-page":"1160112","DOI":"10.3389\/fneur.2023.1160112","article-title":"Exploring the causes of augmentation in restless legs syndrome","volume":"14","author":"Zeng","year":"2023","journal-title":"Front Neurol"},{"key":"ref10","doi-asserted-by":"publisher","first-page":"64","DOI":"10.1016\/j.biopsych.2019.10.018","article-title":"The mesolimbic dopamine system in chronic pain and associated affective comorbidities","volume":"87","author":"Serafini","year":"2020","journal-title":"Biol Psychiatry"},{"key":"ref11","doi-asserted-by":"publisher","first-page":"84","DOI":"10.1016\/j.medcli.2024.05.026","article-title":"Restless legs syndrome. Pathophysiology, diagnosis and treatment","volume":"164","author":"Castillo-Alvarez","year":"2025","journal-title":"Med Clin (Barc)"},{"key":"ref12","doi-asserted-by":"publisher","first-page":"137","DOI":"10.5664\/jcsm.11390","article-title":"Treatment of restless legs syndrome and periodic limb movement disorder: an American Academy of sleep medicine clinical practice guideline","volume":"21","author":"Winkelman","year":"2025","journal-title":"J Clin Sleep Med"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"261","DOI":"10.1002\/ajh.23397","article-title":"The prevalence and impact of restless legs syndrome on patients with iron deficiency anemia","volume":"88","author":"Allen","year":"2013","journal-title":"Am J Hematol"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"114158","DOI":"10.1016\/j.expneurol.2022.114158","article-title":"Brain-iron deficiency models of restless legs syndrome","volume":"356","author":"Earley","year":"2022","journal-title":"Exp Neurol"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"1309","DOI":"10.1007\/s11325-021-02477-y","article-title":"The relationship between sleep disorders, anxiety, depression, and cognitive function with restless legs syndrome (rls) in the elderly","volume":"26","author":"Xu","year":"2022","journal-title":"Sleep Breath"},{"key":"ref16","doi-asserted-by":"publisher","first-page":"82","DOI":"10.1016\/j.sleep.2024.12.004","article-title":"The impact of neuropsychiatric burden on restless legs syndrome (rls) disease severity","volume":"126","author":"Kilincarslan","year":"2025","journal-title":"Sleep Med"},{"key":"ref17","doi-asserted-by":"publisher","first-page":"459","DOI":"10.1097\/AJP.0000000000000133","article-title":"Painful restless legs syndrome: a severe, burning form of the disease","volume":"31","author":"Karroum","year":"2015","journal-title":"Clin J Pain"},{"key":"ref18","doi-asserted-by":"publisher","first-page":"1013","DOI":"10.1001\/archneur.60.7.1013","article-title":"Arm restlessness as the initial symptom in restless legs syndrome","volume":"60","author":"Freedom","year":"2003","journal-title":"Arch Neurol"},{"key":"ref19","doi-asserted-by":"publisher","first-page":"e244890","DOI":"10.1136\/bcr-2021-244890","article-title":"Restless arm syndrome: a rare disease?","volume":"14","author":"Moser","year":"2021","journal-title":"BMJ Case Rep"},{"key":"ref20","doi-asserted-by":"publisher","first-page":"e12258","DOI":"10.1016\/j.heliyon.2022.e12258","article-title":"Restless arms syndrome with oral olanzapine: case based review","volume":"8","author":"Kuang","year":"2022","journal-title":"Heliyon"},{"key":"ref21","doi-asserted-by":"publisher","first-page":"453","DOI":"10.1186\/s12888-021-03433-6","article-title":"Transitory restless arms syndrome in a patient with antipsychotics and antidepressants: a case report","volume":"21","author":"Chen","year":"2021","journal-title":"BMC Psychiatry"},{"key":"ref22","doi-asserted-by":"publisher","first-page":"349","DOI":"10.1186\/s12888-024-05763-7","article-title":"Suspected duloxetine-induced restless legs syndrome phenotypic variant: a case report","volume":"24","author":"Shao","year":"2024","journal-title":"BMC Psychiatry"},{"key":"ref23","doi-asserted-by":"publisher","first-page":"1459","DOI":"10.1002\/mds.25154","article-title":"Characterization of periodic upper limb movement disorder in a patient with restless arms syndrome","volume":"27","author":"Ruppert","year":"2012","journal-title":"Mov Disord"},{"key":"ref24","doi-asserted-by":"publisher","first-page":"1893","DOI":"10.1056\/NEJMcpc2300895","article-title":"Case 15-2023: a 33-year-old man with paresthesia of the arms and legs","volume":"388","author":"Schmitt","year":"2023","journal-title":"N Engl J Med"},{"key":"ref25","doi-asserted-by":"publisher","first-page":"89","DOI":"10.1097\/JCP.0b013e31819302a0","article-title":"Olanzapine-induced \"restless arms syndrome\"","volume":"29","author":"Konstantakopoulos","year":"2009","journal-title":"J Clin Psychopharmacol"},{"key":"ref26","doi-asserted-by":"publisher","first-page":"530","DOI":"10.1016\/S0140-6736(08)60240-8","article-title":"Restless arms","volume":"371","author":"Horvath","year":"2008","journal-title":"Lancet"},{"key":"ref27","doi-asserted-by":"publisher","first-page":"234","DOI":"10.1016\/j.neurol.2016.12.035","article-title":"Pramipexole-responsive acute restless arms syndrome after surgery under general anesthesia: case report and literature review","volume":"173","author":"Donde","year":"2017","journal-title":"Rev Neurol (Paris)"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1725747\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T05:19:36Z","timestamp":1767676776000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1725747\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,6]]},"references-count":27,"alternative-id":["10.3389\/fmed.2025.1725747"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2025.1725747","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,6]]},"article-number":"1725747"}}