{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T14:07:16Z","timestamp":1788444436843,"version":"build-2803163510"},"reference-count":37,"publisher":"MDPI AG","issue":"2","license":[{"start":{"date-parts":[[2023,2,5]],"date-time":"2023-02-05T00:00:00Z","timestamp":1675555200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Ministry of Science and Higher Education of the Russian Federation","award":["075-15-2022-303"],"award-info":[{"award-number":["075-15-2022-303"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Life"],"abstract":"<jats:p>Spinal muscular atrophy (SMA) is characterized by the degeneration of spinal alpha motorneurons. Nusinersen demonstrated good efficacy in the early disease phases. The feasibility of transcutaneous spinal cord stimulation (tSCS) in motor rehabilitation of patients with spinal cord injury has been demonstrated. We hypothesize that tSCS may activate intact and restored by nusinersen motorneurons and slow down the decline in motor activity, and may contribute to the development of motor skills in children with SMA. A case series is presented. Five children (6\u201313 years old) with SMA type II or III participated in the study. They were treated with nusinersen for ~2 years. Application of tSCS was carried out during physical therapy for 30\u201340 min per day in the course of 10\u201314 days. Outcome measures were goniometry of joints with contracture, forced vital capacity (FVC), RULM and HFMSE scales. The participants tolerated the stimulation well. The reduction of the contracture was \u22655 deg. RULM and HFMSE increased by ~1\u20132 points. Predicted FVC increased by 1\u20137% in three participants. Each participant expanded their range of active movements and\/or learned new motor skills. Spinal cord stimulation may be an effective rehabilitation method in patients treated with nusinersen. More research is needed.<\/jats:p>","DOI":"10.3390\/life13020449","type":"journal-article","created":{"date-parts":[[2023,2,6]],"date-time":"2023-02-06T04:08:07Z","timestamp":1675656487000},"page":"449","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["First Use of Non-Invasive Spinal Cord Stimulation in Motor Rehabilitation of Children with Spinal Muscular Atrophy"],"prefix":"10.3390","volume":"13","author":[{"given":"Anton","family":"Novikov","sequence":"first","affiliation":[{"name":"EirMED, 10 Vsevolod Vishnevsky St., 197136 St. Petersburg, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"Maldova","sequence":"additional","affiliation":[{"name":"EirMED, 10 Vsevolod Vishnevsky St., 197136 St. Petersburg, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natalia","family":"Shandybina","sequence":"additional","affiliation":[{"name":"EirMED, 10 Vsevolod Vishnevsky St., 197136 St. Petersburg, Russia"},{"name":"Pavlov Institute of Physiology, Russian Academy of Sciences, 6 Makarova Enb., 199034 St. Petersburg, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ivan","family":"Shalmiev","sequence":"additional","affiliation":[{"name":"EirMED, 10 Vsevolod Vishnevsky St., 197136 St. Petersburg, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elena","family":"Shoshina","sequence":"additional","affiliation":[{"name":"EirMED, 10 Vsevolod Vishnevsky St., 197136 St. Petersburg, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natalia","family":"Epoyan","sequence":"additional","affiliation":[{"name":"EirMED, 10 Vsevolod Vishnevsky St., 197136 St. Petersburg, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8934-5253","authenticated-orcid":false,"given":"Tatiana","family":"Moshonkina","sequence":"additional","affiliation":[{"name":"Pavlov Institute of Physiology, Russian Academy of Sciences, 6 Makarova Enb., 199034 St. Petersburg, Russia"},{"name":"Almazov National Medical Research Centre of the Ministry of Health of the Russian Federation, 2 Akkuratova St., 197341 St. Petersburg, Russia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2023,2,5]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"124","DOI":"10.1186\/s13023-017-0671-8","article-title":"Prevalence, Incidence and Carrier Frequency of 5q\u2013Linked Spinal Muscular Atrophy\u2014A Literature Review","volume":"12","author":"Verhaart","year":"2017","journal-title":"Orphanet J. Rare Dis."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"2120","DOI":"10.1016\/S0140-6736(08)60921-6","article-title":"Spinal Muscular Atrophy","volume":"371","author":"Lunn","year":"2008","journal-title":"Lancet"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"103","DOI":"10.1016\/j.nmd.2017.11.005","article-title":"Diagnosis and Management of Spinal Muscular Atrophy: Part 1: Recommendations for Diagnosis, Rehabilitation, Orthopedic and Nutritional Care","volume":"28","author":"Mercuri","year":"2018","journal-title":"Neuromuscul. Disord."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"1153","DOI":"10.2147\/TCRM.S172291","article-title":"Clinical Evidence Supporting Early Treatment Of Patients With Spinal Muscular Atrophy: Current Perspectives","volume":"15","author":"Dangouloff","year":"2019","journal-title":"Ther. Clin. Risk Manag."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"197","DOI":"10.1016\/j.nmd.2017.11.004","article-title":"Diagnosis and Management of Spinal Muscular Atrophy: Part 2: Pulmonary and Acute Care; Medications, Supplements and Immunizations; Other Organ Systems; and Ethics","volume":"28","author":"Finkel","year":"2018","journal-title":"Neuromuscul. Disord."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"1968","DOI":"10.1089\/neu.2015.4008","article-title":"Noninvasive Reactivation of Motor Descending Control after Paralysis","volume":"32","author":"Gerasimenko","year":"2015","journal-title":"J. Neurotrauma"},{"key":"ref_7","first-page":"350","article-title":"\u0427\u0440\u0435\u0441\u043ao\u0436\u043d\u0430\u044f \u044d\u043b\u0435\u043a\u0442\u0440\u0438\u0447\u0435\u0441\u043a\u0430\u044f \u0441\u0442\u0438\u043c\u0443\u043b\u044f\u0446\u0438\u044f \u0441\u043f\u0438\u043d\u043do\u0433o \u043co\u0437\u0433\u0430 \u0432 \u0434\u0432\u0438\u0433\u0430\u0442\u0435\u043b\u044c\u043do\u0439 \u0440\u0435\u0430\u0431\u0438\u043b\u0438\u0442\u0430\u0446\u0438\u0438 \u043f\u0430\u0446\u0438\u0435\u043d\u0442o\u0432 \u0441 \u0442\u0440\u0430\u0432\u043co\u0439 \u0441\u043f\u0438\u043d\u043do\u0433o \u043co\u0437\u0433\u0430","volume":"1","author":"Moshonkina","year":"2020","journal-title":"\u0418\u043d\u0442\u0435\u0433\u0440\u0430\u0442\u0438\u0432\u043d\u0430\u044f \u0424\u0438\u0437\u0438o\u043bo\u0433\u0438\u044f"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"79","DOI":"10.18019\/1028-4427-2020-26-1-79-88","article-title":"Comprehensive Treatment of a Patient with Complicated Thoracic Spine Injury Using Percutaneous Electrical Spinal Cord Stimulation (Case Report)","volume":"26","author":"Baindurashvili","year":"2020","journal-title":"Genij Ortop."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"5850","DOI":"10.1038\/s41467-021-26026-z","article-title":"Noninvasive Spinal Stimulation Safely Enables Upright Posture in Children with Spinal Cord Injury","volume":"12","author":"Keller","year":"2021","journal-title":"Nat. Commun."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1177\/1545968319893298","article-title":"Transcutaneous Spinal Cord Stimulation and Motor Rehabilitation in Spinal Cord Injury: A Systematic Review","volume":"34","author":"Taylor","year":"2020","journal-title":"Neurorehabil. Neural Repair"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"192","DOI":"10.1016\/j.neulet.2017.01.003","article-title":"Effects of Spinal Cord Stimulation on Motor Functions in Children with Cerebral Palsy","volume":"639","author":"Solopova","year":"2017","journal-title":"Neurosci. Lett."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"5660","DOI":"10.1038\/s41467-022-33208-w","article-title":"A Pilot Study Combining Noninvasive Spinal Neuromodulation and Activity-Based Neurorehabilitation Therapy in Children with Cerebral Palsy","volume":"13","author":"Hastings","year":"2022","journal-title":"Nat. Commun."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"88","DOI":"10.1152\/jn.00084.2018","article-title":"Muscle Coactivation: Definitions, Mechanisms, and Functions","volume":"120","author":"Latash","year":"2018","journal-title":"J. Neurophysiol."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"225","DOI":"10.1016\/j.rehab.2015.05.003","article-title":"Transcutaneous Electrical Spinal-Cord Stimulation in Humans","volume":"58","author":"Gerasimenko","year":"2015","journal-title":"Ann. Phys. Rehabil. Med."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"834","DOI":"10.1152\/jn.00609.2014","article-title":"Initiation and Modulation of Locomotor Circuitry Output with Multisite Transcutaneous Electrical Stimulation of the Spinal Cord in Noninjured Humans","volume":"113","author":"Gerasimenko","year":"2015","journal-title":"J. Neurophysiol."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"e14397","DOI":"10.14814\/phy2.14397","article-title":"The Relationship between Maximum Tolerance and Motor Activation during Transcutaneous Spinal Stimulation Is Unaffected by the Carrier Frequency or Vibration","volume":"8","author":"Manson","year":"2020","journal-title":"Physiol. Rep."},{"key":"ref_17","unstructured":"(2022, November 27). High-Frequency Stimulation Does Not Improve Comfort of Transcutaneous Spinal Cord Stimulation|BioRxiv. Available online: https:\/\/www.biorxiv.org\/content\/10.1101\/2022.09.06.506783v1."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"2633","DOI":"10.1523\/JNEUROSCI.2374-19.2020","article-title":"Cortical and Subcortical Effects of Transcutaneous Spinal Cord Stimulation in Humans with Tetraplegia","volume":"40","author":"Benavides","year":"2020","journal-title":"J. Neurosci."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"78","DOI":"10.1136\/jnnp-2020-324254","article-title":"Motor Unit Changes in Children with Symptomatic Spinal Muscular Atrophy Treated with Nusinersen","volume":"92","author":"Kariyawasam","year":"2021","journal-title":"J. Neurol. Neurosurg. Psychiatry"},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"352","DOI":"10.1177\/0269215508101741","article-title":"Writing SMART Rehabilitation Goals and Achieving Goal Attainment Scaling: A Practical Guide","volume":"23","author":"Botell","year":"2009","journal-title":"Clin. Rehabil."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"2145","DOI":"10.1089\/neu.2017.5461","article-title":"Non-Invasive Activation of Cervical Spinal Networks after Severe Paralysis","volume":"35","author":"Gad","year":"2018","journal-title":"J. Neurotrauma"},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"7733","DOI":"10.1038\/s41598-022-11306-5","article-title":"OPEN Minimal Handgrip Force Is Needed for Transcutaneous Electrical Stimulation to Improve Hand Functions of Patients with Severe Spinal Cord Injury","volume":"12","author":"Huang","year":"2022","journal-title":"Sci. Rep."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"1445","DOI":"10.1001\/archneur.59.9.1445","article-title":"Outcome Measures for Pediatric Spinal Muscular Atrophy","volume":"59","author":"Iannaccone","year":"2002","journal-title":"Arch. Neurol."},{"key":"ref_24","unstructured":"Norkin, C.C., and White, D.J. (2004). Measurement of Joint Motion: A Guide to Goniometry, F.A. Davis Company. [3rd ed.]."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"869","DOI":"10.1002\/mus.25430","article-title":"Revised Upper Limb Module for Spinal Muscular Atrophy: Development of a New Module","volume":"55","author":"Mazzone","year":"2017","journal-title":"Muscle Nerve"},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"693","DOI":"10.1016\/j.nmd.2007.05.009","article-title":"An Expanded Version of the Hammersmith Functional Motor Scale for SMA II and III Patients","volume":"17","author":"Glanzman","year":"2007","journal-title":"Neuromuscul. Disord."},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"426","DOI":"10.1002\/mus.26419","article-title":"Revised Upper Limb Module for Spinal Muscular Atrophy: 12 Month Changes","volume":"59","author":"Pera","year":"2019","journal-title":"Muscle Nerve"},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"e70","DOI":"10.1164\/rccm.201908-1590ST","article-title":"Standardization of Spirometry 2019 Update. An Official American Thoracic Society and European Respiratory Society Technical Statement","volume":"200","author":"Graham","year":"2019","journal-title":"Am. J. Respir. Crit. Care Med."},{"key":"ref_29","doi-asserted-by":"crossref","unstructured":"Chabanon, A., Seferian, A.M., Daron, A., P\u00e9r\u00e9on, Y., Cances, C., Vuillerot, C., De Waele, L., Cuisset, J.-M., Laugel, V., and Schara, U. (2018). Prospective and Longitudinal Natural History Study of Patients with Type 2 and 3 Spinal Muscular Atrophy: Baseline Data NatHis-SMA Study. PLoS ONE, 13.","DOI":"10.1371\/journal.pone.0201004"},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"590","DOI":"10.1097\/PHM.0000000000001869","article-title":"Systematic Review of Motor Function Scales and Patient-Reported Outcomes in Spinal Muscular Atrophy","volume":"101","author":"Wu","year":"2022","journal-title":"Am. J. Phys. Med. Rehabil."},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"3337","DOI":"10.1111\/ene.15501","article-title":"Nusinersen in Adult Patients with 5q Spinal Muscular Atrophy: A Multicenter Observational Cohorts\u2019 Study","volume":"29","author":"Povedano","year":"2022","journal-title":"Eur. J. Neurol."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"625","DOI":"10.1056\/NEJMoa1710504","article-title":"Nusinersen versus Sham Control in Later-Onset Spinal Muscular Atrophy","volume":"378","author":"Mercuri","year":"2018","journal-title":"N. Engl. J. Med."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"1333","DOI":"10.1038\/nn.2401","article-title":"Transformation of Nonfunctional Spinal Circuits into Functional States after the Loss of Brain Input","volume":"12","author":"Courtine","year":"2009","journal-title":"Nat. Neurosci."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"7370","DOI":"10.1523\/JNEUROSCI.1881-08.2008","article-title":"Step Training Reinforces Specific Spinal Locomotor Circuitry in Adult Spinal Rats","volume":"28","author":"Ichiyama","year":"2008","journal-title":"J. Neurosci."},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"74","DOI":"10.3389\/fnmol.2020.00074","article-title":"Motoneuronal Spinal Circuits in Degenerative Motoneuron Disease","volume":"13","author":"Falgairolle","year":"2020","journal-title":"Front. Mol. Neurosci."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"6368","DOI":"10.4081\/ejtm.2017.6368","article-title":"The Contribution of Neuromuscular Stimulation in Elucidating Muscle Plasticity Revisited","volume":"27","author":"Pette","year":"2017","journal-title":"Eur. J. Transl. Myol."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"21","DOI":"10.2106\/JBJS.H.01573","article-title":"How to Design a Good Case Series","volume":"91","author":"Kooistra","year":"2009","journal-title":"J. Bone Jt. Surg."}],"container-title":["Life"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-1729\/13\/2\/449\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T18:24:51Z","timestamp":1760120691000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-1729\/13\/2\/449"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,2,5]]},"references-count":37,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2023,2]]}},"alternative-id":["life13020449"],"URL":"https:\/\/doi.org\/10.3390\/life13020449","relation":{},"ISSN":["2075-1729"],"issn-type":[{"value":"2075-1729","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,2,5]]}}}