{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T04:37:29Z","timestamp":1787027849923,"version":"build-2736575974"},"reference-count":49,"publisher":"MDPI AG","issue":"5","license":[{"start":{"date-parts":[[2023,4,24]],"date-time":"2023-04-24T00:00:00Z","timestamp":1682294400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Children"],"abstract":"<jats:p>Arthrogryposis multiplex congenita is a condition characterised by contractures and deformity in two or more body areas. Physiotherapy may be an appropriate treatment. The aim was to systematically review the evidence for rehabilitation in arthrogryposis multiplex congenita. A systematic review was performed following the PRISMA 2020 criteria. The search was conducted in PubMed, ScienceDirect, Scielo, Scopus, Web of Science, ENFISPO, JSTOR, Google Scholar, ProQuest, Cochrane Library and PEDro from inception until October 2022. To assess the methodological quality, we used the different aspects of the critical appraisal tool JBI. We included 14 studies (6 case reports, 5 case series, 2 cross-sectional and 1 qualitative study). Sample sizes ranged from 1 to 50 participants, with an age range between 11 days and 35 years. Most studies employed multicomponent therapy, mainly kinesitherapy, massage therapy, use of physical agents and stretching, some combined it with orthopaedic therapy, or it was complementary to surgical treatment. The key to improving the clinical picture was early and individualised care, tailored to the characteristics of the patients. Regarding methodological quality, the main conflicts encountered were in the reporting of participant characteristics and experimental interventions. Rehabilitation provides satisfactory results in the treatment of arthrogryposis multiplex congenita. More scientific production and randomised clinical trials are needed.<\/jats:p>","DOI":"10.3390\/children10050768","type":"journal-article","created":{"date-parts":[[2023,4,24]],"date-time":"2023-04-24T03:38:07Z","timestamp":1682307487000},"page":"768","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Rehabilitation in Patients Diagnosed with Arthrogryposis Multiplex Congenita: A Systematic Review"],"prefix":"10.3390","volume":"10","author":[{"given":"Catalina E.","family":"Garc\u00eda Aguilar","sequence":"first","affiliation":[{"name":"Hospital Virgen de las Monta\u00f1as, C. P.\u00ba Ambulatorio, Villamartin, 11650 Cadiz, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2621-2098","authenticated-orcid":false,"given":"Cristina","family":"Garc\u00eda-Mu\u00f1oz","sequence":"additional","affiliation":[{"name":"Department of Nursing and Physiotherapy, University of Cadiz, 11009 Cadiz, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6496-4683","authenticated-orcid":false,"given":"Ines","family":"Carmona-Barrientos","sequence":"additional","affiliation":[{"name":"Department of Nursing and Physiotherapy, University of Cadiz, 11009 Cadiz, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4589-4442","authenticated-orcid":false,"given":"Maria Jesus","family":"Vinolo-Gil","sequence":"additional","affiliation":[{"name":"Department of Nursing and Physiotherapy, University of Cadiz, 11009 Cadiz, Spain"},{"name":"Rehabilitation Clinical Management Unit, Interlevels-Intercenters Hospital Puerta del Mar, Hospital Puerto Real, Cadiz Bay-La Janda Health District, 11006 Cadiz, Spain"},{"name":"Department Biomedical Research and Innovation Institute of Cadiz (INiBICA), Research Unit, Puerta del Mar University Hospital, University of Cadiz, 11009 Cadiz, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4267-9958","authenticated-orcid":false,"given":"Francisco Javier","family":"Martin-Vega","sequence":"additional","affiliation":[{"name":"Department of Nursing and Physiotherapy, University of Cadiz, 11009 Cadiz, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5850-3055","authenticated-orcid":false,"given":"Gloria","family":"Gonzalez-Medina","sequence":"additional","affiliation":[{"name":"Department of Nursing and Physiotherapy, University of Cadiz, 11009 Cadiz, Spain"},{"name":"Department Biomedical Research and Innovation Institute of Cadiz (INiBICA), Research Unit, Puerta del Mar University Hospital, University of Cadiz, 11009 Cadiz, Spain"},{"name":"CTS-986 Physical Therapy and Health (FISA), University Institute of Research in Social Sustainable Development (INDESS), 11009 Cadiz, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2023,4,24]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"e299","DOI":"10.3928\/19382359-20200624-01","article-title":"Arthrogryposis Multiplex Congenita","volume":"49","author":"Langston","year":"2020","journal-title":"Pediatr. Ann."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"288","DOI":"10.1002\/ajmg.c.31721","article-title":"International Multidisciplinary Collaboration toward an Annotated Definition of Arthrogryposis Multiplex Congenita","volume":"181","author":"Cachecho","year":"2019","journal-title":"Am. J. Med. Genet. Part C Semin. Med. Genet."},{"key":"ref_3","doi-asserted-by":"crossref","unstructured":"Kulesa-Mrowiecka, M., Piech, J., Dowgierd, K., and My\u015bliwiec, A. (CRANIO, 2021). Physical Therapy of Temporomandibular Disorder in a Child with Arthrogryposis Multiplex Congenita: A Case Report and Literature Review, CRANIO, ahead of print.","DOI":"10.1080\/08869634.2021.1890453"},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"410","DOI":"10.1002\/ajmg.c.31726","article-title":"Muscle and Joint Function in Children Living with Arthrogryposis Multiplex Congenita: A Scoping Review","volume":"181","author":"Gagnon","year":"2019","journal-title":"Am. J. Med. Genet. Part C Semin. Med. Genet."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"n71","DOI":"10.1136\/bmj.n71","article-title":"The PRISMA 2020 statement: An Updated Guidelinefor Reporting Systematic Reviews","volume":"372","author":"Page","year":"2021","journal-title":"BMJ"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1097\/XEB.0000000000000155","article-title":"The Updated Joanna Briggs Institute Model of Evidence-Based Healthcare","volume":"17","author":"Jordan","year":"2019","journal-title":"Int. J. Evid. Based. Healthc."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"388","DOI":"10.1177\/0309364614541458","article-title":"One-Year Follow-up Study of Serial Orthotic Treatment in Two Cases with Arthrogrypotic Syndromes Who Have Bilateral Knee Flexion Contractures","volume":"40","author":"Erel","year":"2016","journal-title":"Prosthet. Orthot. Int."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"293","DOI":"10.1097\/PEP.0000000000000148","article-title":"A Case Report of an Infant With","volume":"27","author":"Azbell","year":"2015","journal-title":"Pediatr. Phys. Ther."},{"key":"ref_9","first-page":"91","article-title":"Proceso Rehabilitador de Artrogriposis M\u00faltiple Cong\u00e9nita","volume":"27","year":"2013","journal-title":"Rev. Cuba. Ortop. Y Traumatol."},{"key":"ref_10","doi-asserted-by":"crossref","unstructured":"Binkiewicz-Glinska, A., Sobierajska-Rek, A., Bakula, S., Wierzba, J., Drewek, K., Kowalski, I.M., and Zaborowska-Sapeta, K. (2013). Arthrogryposis in Infancy, Multidisciplinary Approach: Case Report. BMC Pediatr., 13.","DOI":"10.1186\/1471-2431-13-184"},{"key":"ref_11","first-page":"118","article-title":"Fisioterapia En Artrogriposis M\u00faltiple Cong\u00e9nita: Caso Cl\u00ednico","volume":"40","year":"2011","journal-title":"Cuest. Fisioter."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"431","DOI":"10.1097\/PHM.0b013e3181a0e249","article-title":"Rehabilitation of an Adult Patient with Arthrogryposis Multiplex Congenita Treated with an External Fixator","volume":"88","author":"Taricco","year":"2009","journal-title":"Am. J. Phys. Med. Rehabil."},{"key":"ref_13","first-page":"751","article-title":"Descripci\u00f3n de Pacientes Con Artrogriposis Cong\u00e9nita En Un Centro de Fisioterapia Pedi\u00e1trica En El Norte de M\u00e9xico","volume":"73","year":"2016","journal-title":"Rev. M\u00e9d. Costa Rica Y Centroam."},{"key":"ref_14","first-page":"27","article-title":"Artrogriposis M\u00faltiple Cong\u00e9nita","volume":"31","author":"Cerisola","year":"2015","journal-title":"Rev. M\u00e9d. Del Urug."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"830","DOI":"10.1016\/j.arcped.2015.05.014","article-title":"L\u2019arthrogrypose: Manifestations Cliniques et Prise En Charge","volume":"22","author":"Ayadi","year":"2015","journal-title":"Arch. Pediatr."},{"key":"ref_16","first-page":"22","article-title":"Results of the Ponseti Method in Patients with Clubfoot Associated with Arthrogryposis","volume":"28","author":"Morcuende","year":"2008","journal-title":"Iowa Orthop. J."},{"key":"ref_17","first-page":"217","article-title":"Enfoque Terap\u00e9utico de La Artrogriposis","volume":"26","year":"1992","journal-title":"Rehabilitation"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1016\/j.jped.2015.04.008","article-title":"Characterization of a Group Unrelated Patients with Arthrogryposis Multiplex Congenita","volume":"92","author":"Kofman","year":"2016","journal-title":"J. Pediatr."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"214","DOI":"10.1097\/BPO.0b013e3181990214","article-title":"Ambulatory Activity in Youth with Arthrogryposis: A Cohort Study","volume":"29","author":"Dillon","year":"2009","journal-title":"J. Pediatr. Orthop."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"2318","DOI":"10.1080\/09638288.2018.1559364","article-title":"Rehabilitation Needs of Youth with Arthrogryposis Multiplex Congenita: Perspectives from Key Stakeholders","volume":"42","author":"Elfassy","year":"2020","journal-title":"Disabil. Rehabil."},{"key":"ref_21","first-page":"1","article-title":"Protocolo de Atenci\u00f3n Kin\u00e9sica En Ni\u00f1os Con Artrogriposis M\u00faltiple Cong\u00e9nita","volume":"86","author":"Melina","year":"2003","journal-title":"Univ. Abierta Interam."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1016\/j.jevs.2018.02.003","article-title":"Gait and Gross Motor Improvements in a Two-Year-Old Child With Arthrogryposis After Hippotherapy Intervention Using a Norwegian Fjord","volume":"67","author":"Brady","year":"2018","journal-title":"J. Equine Vet. Sci."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"399","DOI":"10.15448\/1980-6108.2014.4.17752","article-title":"Atua\u00e7\u00e3o Da Fisioterapia Aqu\u00e1tica Funcional No Deslocamento Na Postura Sentada Na Amioplasia Cong\u00eanita: Relato de Caso","volume":"24","author":"Hengles","year":"2015","journal-title":"Sci. Med."},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"436","DOI":"10.1002\/ajmg.c.31725","article-title":"Pain among Children and Adults Living with Arthrogryposis Multiplex Congenita: A Scoping Review","volume":"181","author":"Cirillo","year":"2019","journal-title":"Am. J. Med. Genet. Part C Semin. Med. Genet."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"e18688","DOI":"10.2196\/18688","article-title":"A Telerehabilitation Intervention for Youths With Arthrogryposis Multiplex Congenita: Protocol for a Pilot Study","volume":"9","author":"Gagnon","year":"2020","journal-title":"JMIR Res. Protoc."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"281","DOI":"10.1007\/s00132-007-1044-0","article-title":"Arthrogryposis Multiplex Congenita","volume":"36","author":"Parsch","year":"2007","journal-title":"Orthopade"},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"48","DOI":"10.1007\/s11684-017-0500-4","article-title":"Arthrogryposis Multiplex Congenita: Classification, Diagnosis, Perioperative Care, and Anesthesia","volume":"11","author":"Ma","year":"2017","journal-title":"Front. Med."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"44","DOI":"10.18233\/APM40No1pp44-501761","article-title":"Abordaje Cl\u00ednico y Diagn\u00f3stico de La Artrogriposis","volume":"40","year":"2019","journal-title":"Acta Pedi\u00e1trica M\u00e9xico"},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"e424","DOI":"10.1016\/j.wneu.2021.10.115","article-title":"Improvement of Pulmonary Function in Arthrogryposis Multiplex Congenita Patients Undergoing Posterior Spinal Fusion Surgery for Concomitant Scoliosis: A Minimum of 3-Year Follow-Up","volume":"157","author":"Xu","year":"2022","journal-title":"World Neurosurg."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"E600","DOI":"10.1097\/BRS.0000000000003347","article-title":"Proximal Junctional Kyphosis After Posterior Spinal Fusion for Severe Kyphoscoliosis in a Patient With PIEZO2-Deficient Arthrogryposis Syndrome","volume":"45","author":"Uehara","year":"2020","journal-title":"Spine"},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"908","DOI":"10.1213\/ANE.0000000000001822","article-title":"Anesthetic Outcomes of Children with Arthrogryposis Syndromes: No Evidence of Hyperthermia","volume":"124","author":"Gleich","year":"2017","journal-title":"Anesth. Analg."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1016\/j.jpeds.2019.08.022","article-title":"50 Years Ago in THE JOURNAL OF PEDIATRICS: Arthrogryposis Multiplex Congenita: A Clinical Investigation","volume":"217","author":"Giampietro","year":"2020","journal-title":"J. Pediatr."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"118","DOI":"10.1016\/j.jpeds.2019.03.006","article-title":"The Role of Patient and Parental Resilience in Adolescents with Chronic Musculoskeletal Pain","volume":"210","author":"Gmuca","year":"2019","journal-title":"J. Pediatr."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1007\/s40271-018-0336-2","article-title":"Parent Empowerment in Pediatric Healthcare Settings: A Systematic Review of Observational Studies","volume":"12","author":"Ashcraft","year":"2019","journal-title":"Patient-Patient-Cent. Outcomes Res."},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"105223","DOI":"10.1016\/j.earlhumdev.2020.105223","article-title":"Effectiveness of a Home-Based Early Cognitive-Motor Intervention Provided in Daycare, Home Care, and Foster Care Settings: Changes in Motor Development and Context Affordances","volume":"151","author":"Valentini","year":"2020","journal-title":"Early Hum. Dev."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1016\/j.jadohealth.2018.11.014","article-title":"Changes in Health-Related Outcomes in Youth With Obesity in Response to a Home-Based Parent-Led Physical Activity Program","volume":"65","author":"Rubin","year":"2019","journal-title":"J. Adolesc. Heal."},{"key":"ref_37","first-page":"81","article-title":"Validaci\u00f3n de La PDMS-2 En Poblaci\u00f3n Espa\u00f1ola. Evaluaci\u00f3n de La Intervenci\u00f3n de Fisioterapia y La Participaci\u00f3n de Los Padres En El Tratamiento de Ni\u00f1os Con Trastornos Del Neurodesarrollo","volume":"73","author":"Caballero","year":"2021","journal-title":"Rev. Neurol."},{"key":"ref_38","doi-asserted-by":"crossref","first-page":"243","DOI":"10.1111\/dmcn.15029","article-title":"Task-Specific Training for Bicycle-Riding Goals in Ambulant Children with Cerebral Palsy: A Randomized Controlled Trial","volume":"64","author":"Toovey","year":"2022","journal-title":"Dev. Med. Child Neurol."},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"105488","DOI":"10.1016\/j.earlhumdev.2021.105488","article-title":"General Movement Optimality Score and General Movements Trajectories Following Early Parent-Administrated Physiotherapy in the Neonatal Intensive Care Unit","volume":"163","author":"Ustad","year":"2021","journal-title":"Early Hum. Dev."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"639","DOI":"10.1111\/1460-6984.12542","article-title":"Randomized Controlled Trial Comparing Parent Led Therapist Supervised Articulation Therapy (PLAT) with Routine Intervention for Children with Speech Disorders Associated with Cleft Palate","volume":"55","author":"Sweeney","year":"2020","journal-title":"Int. J. Lang. Commun. Disord."},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1016\/j.apmr.2021.07.807","article-title":"Parent Experience and Cost Savings Associated With a Novel Tele-Physiatry Program for Children Living in Rural and Underserved Communities","volume":"103","author":"Davidson","year":"2022","journal-title":"Arch. Phys. Med. Rehabil."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1080\/17518423.2019.1655675","article-title":"Evaluation of a Web Platform Aiming to Support Parents Having a Child with Developmental Coordination Disorder: Brief Report","volume":"23","author":"Pratte","year":"2020","journal-title":"Dev. Neurorehabil."},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"479","DOI":"10.1093\/tropej\/fmaa001","article-title":"Comparative Effect of Mother\u2019s Hug and Massage on Neonatal Pain Behaviors Caused by Blood Sampling: A Randomized Clinical Trial","volume":"66","author":"Roshanray","year":"2020","journal-title":"J. Trop. Pediatr."},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"1068043","DOI":"10.3389\/fpubh.2022.1068043","article-title":"The Positive Effect of Mother-Performed Infant Massage on Infantile Eczema and Maternal Mental State: A Randomized Controlled Trial","volume":"10","author":"Lin","year":"2023","journal-title":"Front. Public Heal."},{"key":"ref_45","doi-asserted-by":"crossref","first-page":"145","DOI":"10.2739\/kurumemedj.MS663003","article-title":"An Educational Module for Pavlik Harness Application for Developmental Dysplasia of the Hip: Study in a Greek Population","volume":"66","author":"Touzopoulos","year":"2019","journal-title":"Kurume Med. J."},{"key":"ref_46","first-page":"4","article-title":"Evaluation of the Efficiency of the Web-Based Epilepsy Education Program (WEEP) for Youth with Epilepsy and Parents: A Randomized Controlled Trial","volume":"111","author":"Duman","year":"2020","journal-title":"Epilepsy Behav."},{"key":"ref_47","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1111\/jir.12983","article-title":"A Pilot Randomised Controlled Trial on the Effectiveness of Infant Massage on the Acceptance, Commitment and Awareness of Influence in Parents of Babies with Down Syndrome","volume":"67","year":"2023","journal-title":"J. Intellect. Disabil. Res."},{"key":"ref_48","doi-asserted-by":"crossref","first-page":"608","DOI":"10.12998\/wjcc.v2.i11.608","article-title":"Important of Case-Reports\/Series, in Rare Diseases: Using Neuroendocrine Tumors as an Example","volume":"2","author":"Nakamura","year":"2014","journal-title":"World J. Clin. Cases"},{"key":"ref_49","doi-asserted-by":"crossref","unstructured":"Sampayo-Cordero, M., Miguel-Huguet, B., Malfettone, A., P\u00e9rez-Garc\u00eda, J.M., Llombart-Cussac, A., Cort\u00e9s, J., Pardo, A., and P\u00e9rez-L\u00f3pez, J. (2020). The Value of Case Reports in Systematic Reviews from Rare Diseases. The Example of Enzyme Replacement Therapy (ERT) in Patients with Mucopolysaccharidosis Type II (MPS-II). Int. J. Environ. Res. Public Health, 17.","DOI":"10.3390\/ijerph17186590"}],"container-title":["Children"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2227-9067\/10\/5\/768\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T19:22:11Z","timestamp":1760124131000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2227-9067\/10\/5\/768"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,4,24]]},"references-count":49,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2023,5]]}},"alternative-id":["children10050768"],"URL":"https:\/\/doi.org\/10.3390\/children10050768","relation":{},"ISSN":["2227-9067"],"issn-type":[{"value":"2227-9067","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,4,24]]}}}