Literature DB >> 28875772

Atypical caudal regression syndrome with agenesis of lumbar spine and presence of sacrum - case report and literature review.

Edyta Szumera1, Barbara Jasiewicz1, Tomasz Potaczek1.   

Abstract

CONTEXT: Caudal regression syndrome is a rare disorder, not well described in the literature.
FINDINGS: Authors treated two patients with congenital absence of thoracolumbar vertebrae and lower limbs paraplegia. Patients had hypoplasia of the lower trunk and extremities with motion between upper and lower torso. Imaging showed caudal spine agenesis, but cleft sacrum was present. Due to severe kyphotic deformity and spinal instability, deformity correction and posterior fusion was performed at the age 6 and 8. Finally, fusion was achieved in one case and stable but non-fusion kyphotic posture was observed in second.
CONCLUSION: surgery in caudal regression syndrome is challenging and bears high risk of complications.

Entities:  

Keywords:  Caudal regression syndrome; Congenital spinal defect; Lower limb paraplegia; Spinal agenesis; Spine surgery

Mesh:

Year:  2017        PMID: 28875772      PMCID: PMC6055966          DOI: 10.1080/10790268.2017.1369211

Source DB:  PubMed          Journal:  J Spinal Cord Med        ISSN: 1079-0268            Impact factor:   1.985


  17 in total

1.  Lumbopelvic stabilization with external fixator in a patient with lumbosacral agenesis.

Authors:  Jacques Griffet; Julien Leroux; Toni El Hayek
Journal:  Eur Spine J       Date:  2010-05-26       Impact factor: 3.134

2.  Caudal regression syndrome.

Authors:  Dino Samartzis; Francis H Shen
Journal:  Ann Acad Med Singap       Date:  2008-05       Impact factor: 2.473

3.  Presentation and outcome of sacral agenesis: 20 years' experience.

Authors:  J M Wilmshurst; R Kelly; M Borzyskowski
Journal:  Dev Med Child Neurol       Date:  1999-12       Impact factor: 5.449

Review 4.  Caudal regression syndrome: etiopathogenesis, prenatal diagnosis, and perinatal management.

Authors:  A Adra; D Cordero; A Mejides; S Yasin; F Salman; M J O'Sullivan
Journal:  Obstet Gynecol Surv       Date:  1994-07       Impact factor: 2.347

Review 5.  Autosomal dominant sacral agenesis: Currarino syndrome.

Authors:  S A Lynch; Y Wang; T Strachan; J Burn; S Lindsay
Journal:  J Med Genet       Date:  2000-08       Impact factor: 6.318

6.  Kyphectomy in children with myelomeningocele.

Authors:  Haluk Altiok; Craig Finlayson; Sahar Hassani; Peter Sturm
Journal:  Clin Orthop Relat Res       Date:  2011-05       Impact factor: 4.176

7.  Lumbosacral agenesis. Three cases of reconstruction using Cotrel-Dubousset or L-rod instrumentation.

Authors:  C E Dumont; J P Damsin; V Forin; H Carlioz
Journal:  Spine (Phila Pa 1976)       Date:  1993-07       Impact factor: 3.468

8.  Diagnosis and management of sacral agenesis.

Authors:  T Gotoh; Y Shinno; S Kobayashi; Y Watarai; T Koyanagi
Journal:  Eur Urol       Date:  1991       Impact factor: 20.096

9.  Maternal diabetes increases the risk of caudal regression caused by retinoic acid.

Authors:  Billy W H Chan; Kwok-Siu Chan; Tsuyoshi Koide; Sau-Man Yeung; Maran B W Leung; Andrew J Copp; Mary R Loeken; Toshihiko Shiroishi; Alisa S W Shum
Journal:  Diabetes       Date:  2002-09       Impact factor: 9.461

10.  Sirenomelia and severe caudal regression syndrome.

Authors:  Mohammed Z Seidahmed; Omer B Abdelbasit; Khalid A Alhussein; Abeer M Miqdad; Mohammed I Khalil; Mustafa A Salih
Journal:  Saudi Med J       Date:  2014-12       Impact factor: 1.484

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