{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,6]],"date-time":"2026-02-06T04:10:30Z","timestamp":1770351030908,"version":"3.49.0"},"reference-count":44,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"18","license":[{"start":{"date-parts":[[2024,4,29]],"date-time":"2024-04-29T00:00:00Z","timestamp":1714348800000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,4,29]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Lipomyelomeningocele associated with an ulnar club hand in the spectrum of VACTERL association ([costo-]vertebral abnormalities; anal atresia; cardiac defects; tracheal-esophageal abnomalities, including atresia, stenosis, and fistula; renal and radial abnormalities; limb abnormalities; single umbilical artery) is a very rare and infrequently reported phenomenon. Within the fat mass of the lipoma, it is not common to find a well-defined cartilaginous mass with no attachments to the surrounding tissue.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>The authors present the case of a 3-month-old male with low-back swelling that was off-center to the left, accompanied by a left short forearm displaying outward bowing. Echocardiography showed an atrial septal defect. This rare VACTERL association comprises lipomyelomeningocele, atrial septal defect, and ulnar longitudinal deficiency syndrome. During surgical intervention for the lipoma, a well-defined cartilaginous mass was discovered within the adipose tissue.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>The manifestation of VACTERL association can be partially explained by the Shh\/Gli and Wnt pathway defects. It is prudent to screen children with neural tube defects to be aware of any associated syndromes. This case is very rare, and the literature has contained no prior report on the VACTERL association of lipomyelomeningocele, atrial septal defect, and ulnar longitudinal deficiency.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case24177","type":"journal-article","created":{"date-parts":[[2024,4,29]],"date-time":"2024-04-29T02:45:52Z","timestamp":1714358752000},"source":"Crossref","is-referenced-by-count":2,"title":["Cartilage within lipomyelomeningocele and ulnar longitudinal deficiency syndrome as VACTERL association, alliance in SHH\/GLI3, and Wnt pathway: illustrative case"],"prefix":"10.3171","volume":"7","author":[{"given":"Mikael Aseged","family":"Shimekit","sequence":"first","affiliation":[{"name":"Department of Surgery, Division of Neurosurgery, Asrat Woldeyes Health Science Campus, Debre Berhan University, Debre Berhan, Ethiopia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ermias Fikru","family":"Yesuf","sequence":"additional","affiliation":[{"name":"Department of Surgery, Division of Neurosurgery, Asrat Woldeyes Health Science Campus, Debre Berhan University, Debre Berhan, Ethiopia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Simon Mulugeta","family":"Teferi","sequence":"additional","affiliation":[{"name":"Department of Pathology, Asrat Woldeyes Health Science Campus, Debre Berhan University, Debre Berhan, Ethiopia; and"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mahlet Getachew","family":"Lemma","sequence":"additional","affiliation":[{"name":"Department of Public Health, Gamby University, Addis Ababa, Ethiopia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1754","reference":[{"issue":"3","key":"b1","series-title":"J Pediatr","first-page":"458","article-title":"VACTERL (vertebral defects, anal atresia, tracheoesophageal fistula with esophageal atresia, cardiac defects, renal and limb anomalies) association: disease spectrum in 25 patients ascertained for their upper limb involvement","volume":"164","author":"Carli D","year":"2014","unstructured":"Carli D, Garagnani L, Lando M, Fairplay T, Bernasconi S, Landi A,  VACTERL (vertebral defects, anal atresia, tracheoesophageal fistula with esophageal atresia, cardiac defects, renal and limb anomalies) association: disease spectrum in 25 patients ascertained for their upper limb involvement. J Pediatr. 2014;164(3):458\u2013462.e2."},{"issue":"4","key":"b2","series-title":"J Neurosurg Pediatr","first-page":"1","article-title":"External validation of a new classification of spinal lipomas based on embryonic stage","volume":"25","author":"Tominey S","year":"2020","unstructured":"Tominey S, Kaliaperumal C, Gallo P. External validation of a new classification of spinal lipomas based on embryonic stage. J Neurosurg Pediatr. 2020;25(4):1\u20138."},{"issue":"4","key":"b3","series-title":"Neurosurg Focus","first-page":"E3","article-title":"Lipomyelomeningocele: pathology, treatment, and outcomes","volume":"33","author":"Sarris CE","year":"2012","unstructured":"Sarris CE, Tomei KL, Carmel PW, Gandhi CD. Lipomyelomeningocele: pathology, treatment, and outcomes. Neurosurg Focus. 2012;33(4):E3."},{"issue":"12","key":"b4","series-title":"Childs Nerv Syst","first-page":"2123","article-title":"Accessory lower limb associated with spina bifida: case report","volume":"30","author":"Bayri Y","year":"2014","unstructured":"Bayri Y, Tanr\u0131kulu B, Ek\u015fi M\u015e, Da\u011f\u00e7\u0131nar A. Accessory lower limb associated with spina bifida: case report. Childs Nerv Syst. 2014;30(12):2123\u20132126."},{"issue":"1","key":"b5","series-title":"Case Rep Clin Radiol","first-page":"47","article-title":"Anomalous osseous limb: a sneak peek into rare association with lipomyelomeningocele\u2014a case report","volume":"1","author":"Dogra L","year":"2023","unstructured":"Dogra L, Bhayana A, Dakshayini HS, Bagri N. Anomalous osseous limb: a sneak peek into rare association with lipomyelomeningocele\u2014a case report. 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