{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T11:16:40Z","timestamp":1791285400094,"version":"4.1.0"},"reference-count":30,"publisher":"MDPI AG","issue":"2","license":[{"start":{"date-parts":[[2026,1,15]],"date-time":"2026-01-15T00:00:00Z","timestamp":1768435200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100003725","name":"National Research Foundation of Korea","doi-asserted-by":"crossref","award":["RS-2025-16065320"],"award-info":[{"award-number":["RS-2025-16065320"]}],"id":[{"id":"10.13039\/501100003725","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>Background: Hand oligodactyly is a rare congenital malformation characterized by fewer than five digits. Although several clinical and radiological studies have been reported, detailed anatomical investigations remain scarce. This study aimed to describe the morphological characteristics of bilateral hand oligodactyly through dissection. Case Presentation: A bilateral hand oligodactyly with thumb and 3 fingers on each hand was found in a 77-year-old Korean female donor. The study was approved by the Institutional Review Board of Sungkyunkwan University (IRB No. SKKU 2023-02-012). Results: Diverse variations were observed in most structures of the hand, including the palmar aponeurosis, tendons of the flexor digitorum superficialis, the flexor digitorum profundus, and the flexor pollicis longus, lumbricals, interossei, and metacarpals. Connections between flexor and extensor tendons were also found, and each variation differed between the two hands. Conclusions: These structural differences highlight the developmental complexity of hand oligodactyly and offer valuable insights for facilitating appropriate surgical strategies. This study has also provided new insights into congenital anomalies that underscore their relevance in developmental biology.<\/jats:p>","DOI":"10.3390\/diagnostics16020275","type":"journal-article","created":{"date-parts":[[2026,1,15]],"date-time":"2026-01-15T11:13:21Z","timestamp":1768475601000},"page":"275","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Anatomical Variations in Bilateral Hand Oligodactyly: A Case Analysis of Cleft Hand"],"prefix":"10.3390","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1482-1657","authenticated-orcid":false,"given":"Mi-Sun","family":"Hur","sequence":"first","affiliation":[{"name":"Department of Anatomy, Daegu Catholic University School of Medicine, Daegu 42472, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3169-5197","authenticated-orcid":false,"given":"Jae Woo","family":"Shim","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul 06351, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ju-Hyeon","family":"Bae","sequence":"additional","affiliation":[{"name":"Department of Molecular Cell Biology, Sungkyunkwan University School of Medicine, Suwon 16419, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jong-Sun","family":"Kang","sequence":"additional","affiliation":[{"name":"Department of Molecular Cell Biology, Sungkyunkwan University School of Medicine, Suwon 16419, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5380-6815","authenticated-orcid":false,"given":"Kwang Il","family":"Nam","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Chonnam National University Medical School, Gwangju 58128, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7767-931X","authenticated-orcid":false,"given":"Chang-Seok","family":"Oh","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Sungkyunkwan University School of Medicine, 2066, Seobu-Ro, Jangan-Gu, Suwon 6419, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2026,1,15]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"110","DOI":"10.1053\/jssh.2002.34791","article-title":"Fewer than 10: Oligodactyly\u2014Diagnoses and patterns of malformation","volume":"2","author":"Conrad","year":"2002","journal-title":"J. Am. Soc. Surg. Hand"},{"key":"ref_2","unstructured":"Davis, D.D., and Kane, S.M. (2025). Cleft hand. StatPearls, StatPearls Publishing."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"523","DOI":"10.1111\/j.1440-169X.2007.00939.x","article-title":"Clinical features and teratogenic mechanisms of congenital absence of digits","volume":"49","author":"Ogino","year":"2007","journal-title":"Dev. Growth Differ."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"1144","DOI":"10.1038\/ejhg.2011.97","article-title":"17p13.3 microduplications are associated with split-hand\/foot malformation and long-bone deficiency (SHFLD)","volume":"19","author":"Armour","year":"2011","journal-title":"Eur. J. Hum. Genet."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"283","DOI":"10.1142\/S2424835516400087","article-title":"Oligodactyly with thumb","volume":"21","author":"Baek","year":"2016","journal-title":"J. Hand Surg. Asian Pac. Vol."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"1707","DOI":"10.2106\/00004623-196446080-00006","article-title":"Cleft hand: Classification, incidence, and treatment. Review of the literature and report of nineteen cases","volume":"46","author":"Barsky","year":"1964","journal-title":"J. Bone Jt. Surg. Am."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"1579","DOI":"10.1016\/j.jhsa.2008.05.010","article-title":"Central ray deficiency: Subjective and objective outcome of cleft reconstruction","volume":"33","author":"Goldfarb","year":"2008","journal-title":"J. Hand Surg. Am."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1288","DOI":"10.1016\/j.jhsa.2005.07.006","article-title":"Type 0 ulnar longitudinal deficiency","volume":"30","author":"Havenhill","year":"2005","journal-title":"J. Hand Surg. Am."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"132","DOI":"10.1111\/cge.13434","article-title":"Genetic regulatory pathways of split-hand\/foot malformation","volume":"95","author":"Kantaputra","year":"2019","journal-title":"Clin. Genet."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1407","DOI":"10.1016\/j.radcr.2019.09.004","article-title":"Atypical cleft hand with complex syndactyly: An unusual form of hand oligodactyly","volume":"14","author":"Kavak","year":"2019","journal-title":"Radiol. Case Rep."},{"key":"ref_11","doi-asserted-by":"crossref","unstructured":"Paththinige, C.S., Sirisena, N.D., Escande, F., Manouvrier, S., Petit, F., and Dissanayake, V.H.W. (2019). Split hand\/foot malformation with long bone deficiency associated with BHLHA9 gene duplication: A case report and review of the literature. BMC Med. Genet., 20.","DOI":"10.1186\/s12881-019-0839-2"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1007\/s13353-013-0178-5","article-title":"Split-hand\/foot malformation\u2014Molecular cause and implications in genetic counseling","volume":"55","author":"Socha","year":"2014","journal-title":"J. Appl. Genet."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"106","DOI":"10.3109\/03093649109164644","article-title":"The surgical treatment of congenital hand deficiency","volume":"15","author":"Sykes","year":"1991","journal-title":"Prosthet. Orthot. Int."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"658","DOI":"10.1016\/S0363-5023(84)80007-6","article-title":"Ulnar ray deficiency: Its various manifestations","volume":"9","author":"Swanson","year":"1984","journal-title":"J. Hand Surg. Am."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"229","DOI":"10.1007\/BF00269920","article-title":"Central ray deficiency of the hand: Operative treatment and results","volume":"8","author":"Tada","year":"1984","journal-title":"Int. Orthop."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"138","DOI":"10.1159\/000335405","article-title":"Correlation between 3D ultrasound appearance and postnatal findings in bilateral malformations of the fetal hands","volume":"31","author":"Muscatello","year":"2012","journal-title":"Fetal Diagn. Ther."},{"key":"ref_17","doi-asserted-by":"crossref","unstructured":"Bergeron, V., and Kajganic, P. (2024). Pressure sensors for evaluating hand grasp and pinch. Sensors, 24.","DOI":"10.3390\/s24175768"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"361","DOI":"10.1016\/j.hcl.2016.03.006","article-title":"A comprehensive guide on restoring grasp using tendon transfer procedures for ulnar nerve palsy","volume":"32","author":"Chung","year":"2016","journal-title":"Hand Clin."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"9","DOI":"10.1016\/j.hcl.2011.09.005","article-title":"The interosseous muscles: The foundation of hand function","volume":"28","author":"Liss","year":"2012","journal-title":"Hand Clin."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"476","DOI":"10.5435\/JAAOS-D-14-00056","article-title":"Ray resections of the fingers: Indications, techniques, and outcomes","volume":"23","author":"Blazar","year":"2015","journal-title":"J. Am. Acad. Orthop. Surg."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"129","DOI":"10.1016\/j.hcl.2021.11.001","article-title":"Anatomy and biomechanics of the thumb carpometacarpal joint","volume":"38","author":"Nichols","year":"2022","journal-title":"Hand Clin."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"480","DOI":"10.1016\/j.jhsa.2009.12.021","article-title":"Correction of the typical cleft hand","volume":"35","author":"Upton","year":"2010","journal-title":"J. Hand Surg. Am."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"179","DOI":"10.1242\/dev.024158","article-title":"Growing models of vertebrate limb development","volume":"136","author":"Towers","year":"2009","journal-title":"Development"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"845","DOI":"10.1038\/nrg2681","article-title":"Vertebrate limb bud development: Moving towards integrative analysis of organogenesis","volume":"10","author":"Zeller","year":"2009","journal-title":"Nat. Rev. Genet."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"109","DOI":"10.4161\/org.4.2.5857","article-title":"Wnt signaling in limb organogenesis","volume":"4","author":"Nimmagadda","year":"2008","journal-title":"Organogenesis"},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"939","DOI":"10.1016\/0092-8674(95)90297-X","article-title":"Interaction between the signaling molecules Wnt7a and Shh during vertebrate limb development\u2014Dorsal signals regulate anteroposterior patterning","volume":"80","author":"Yang","year":"1995","journal-title":"Cell"},{"key":"ref_27","doi-asserted-by":"crossref","unstructured":"Ahmed, H., Akbari, H., Emami, A., and Akbari, M.R. (2017). Genetic overview of syndactyly and polydactyly. Plast. Reconstr. Surg. Glob. Open, 5.","DOI":"10.1097\/GOX.0000000000001549"},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"459","DOI":"10.1016\/j.devcel.2017.04.021","article-title":"On the formation of digits and joints during limb development","volume":"41","author":"Hiscock","year":"2017","journal-title":"Dev. Cell"},{"key":"ref_29","doi-asserted-by":"crossref","unstructured":"Farin, H.F., L\u00fcdtke, T.H.-W., Schmidt, M.K., Placzko, S., Schuster-Gossler, K., Petry, M., Christoffels, V.M., and Kispert, A. (2013). Tbx2 terminates Shh\/FGF signaling in the developing mouse limb bud by direct repression of Gremlin1. PLoS Genet., 9.","DOI":"10.1371\/journal.pgen.1003467"},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"1303","DOI":"10.1002\/dvdy.22637","article-title":"Analysis of mutants with altered Shh activity and posterior digit loss supports a biphasic model for Shh function as a morphogen and mitogen","volume":"240","author":"Zhu","year":"2011","journal-title":"Dev. Dyn."}],"container-title":["Diagnostics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-4418\/16\/2\/275\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,17]],"date-time":"2026-01-17T05:40:48Z","timestamp":1768628448000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-4418\/16\/2\/275"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,15]]},"references-count":30,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2026,1]]}},"alternative-id":["diagnostics16020275"],"URL":"https:\/\/doi.org\/10.3390\/diagnostics16020275","relation":{},"ISSN":["2075-4418"],"issn-type":[{"value":"2075-4418","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,15]]}}}