title: Sacral agenesis: evaluation of accompanying pathologies in 38 cases, with analysis of long-term outcomes.
abstract: OBJECTIVE
  Sacral agenesis (SA), or caudal regression syndrome, is a congenital malformation of the spine of varying degree of severity. The aim of our study was to identify associated impairments in structure and function of the orthopedic, neurological, cardiopulmonary, genitourinary, and gastrointestinal systems, and to evaluate their impact on function.
  
  
  METHODS
  This was a retrospective case series analysis of 38 patients with SA. Patients were divided into two groups: SA with myelomeningocele (group 1) and without myelomeningocele (group 2). Between-group comparisons in terms of the features of the SA, impairments in associated systems, impact on gross motor function, need for surgery, and association with prenatal screening and maternal gestational diabetes were evaluated.
  
  
  RESULTS
  The majority of comorbidities were orthopedic and neurological in nature. Impairments in sphincter control and independent transferring were more prevalent in group 1, resulting in lower function. Scoliosis, kyphosis, and hip dislocation/subluxation were the most common orthopedic problems, with a higher prevalence of kyphosis in group 1. The requirement for neurosurgery was significantly higher in group 1.
  
  
  CONCLUSIONS
  Orthopedic and neurological comorbidities are commonly associated with SA and are more prevalent in the presence of a myelomeningocele. As the impairments impact a child's ability for maximum function, early identification and intervention is required to correct or ameliorate the impairment.
  
  
  METHODS
  Level IV.
authors.affiliation: Metin Sabanci Baltalimani Bone Diseases Training and Research Hospital, Baltalimani, 34470 Sariyer, Istanbul, Turkey. mbbalibey@gmail.com.
authors.forename: Mehmet Bülent
authors.identifier: http://orcid.org/0000-0001-5127-2004
authors.initials: MB
authors.lastname: Balioğlu
authors.affiliation: Metin Sabanci Baltalimani Bone Diseases Training and Research Hospital, Baltalimani, 34470 Sariyer, Istanbul, Turkey.
authors.forename: Yunus Emre
authors.initials: YE
authors.lastname: Akman
authors.affiliation: Metin Sabanci Baltalimani Bone Diseases Training and Research Hospital, Baltalimani, 34470 Sariyer, Istanbul, Turkey.
authors.forename: Hanifi
authors.initials: H
authors.lastname: Ucpunar
authors.affiliation: Metin Sabanci Baltalimani Bone Diseases Training and Research Hospital, Baltalimani, 34470 Sariyer, Istanbul, Turkey.
authors.forename: Akif
authors.initials: A
authors.lastname: Albayrak
authors.affiliation: Metin Sabanci Baltalimani Bone Diseases Training and Research Hospital, Baltalimani, 34470 Sariyer, Istanbul, Turkey.
authors.forename: Deniz
authors.initials: D
authors.lastname: Kargın
authors.affiliation: Metin Sabanci Baltalimani Bone Diseases Training and Research Hospital, Baltalimani, 34470 Sariyer, Istanbul, Turkey.
authors.forename: Yunus
authors.initials: Y
authors.lastname: Atıcı
authors.affiliation: Dogubayazit State Hospital, Doğubeyazıt, Agrı, Turkey.
authors.forename: Abdül Fettah
authors.initials: AF
authors.lastname: Büyük
mesh_terms: D000015:Abnormalities, Multiple / Q000000981:diagnostic imaging* / Q000601:surgery*; D000293:Adolescent; D002648:Child; D002675:Child, Preschool; D015331:Cohort Studies; D005260:Female; D006801:Humans; D007223:Infant; D008297:Male; D008588:Meningocele / Q000000981:diagnostic imaging* / Q000601:surgery*; D012189:Retrospective Studies; D012445:Sacrococcygeal Region / Q000002:abnormalities* / Q000000981:diagnostic imaging / Q000601:surgery; D013997:Time Factors; D016896:Treatment Outcome; D055815:Young Adult
keywords: Caudal regression syndrome; Congenital anomaly; Sacral agenesis; Spina bifida; Spinal dysraphism