Caudal Duplication Syndrome

Domínguez, Rodrigo; Rott, John; Castillo, Maurício; Pittaluga, Roberto R.; Corriere, Joseph N.

doi:10.1001/archpedi.1993.02160340034009

Cited by 58 publications

(75 citation statements)

References 8 publications

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“…Incesu et al [10] reported another adolescent case of a 15-year-old girl complaining of back pain, and diagnosed asymmetric lumbar spine duplication with spinal cord tethering secondary to a filum lipoma in the sacrum. The other cases of duplication of spine were symptomatic with neurovascular, genitourinary, or gastrointestinal abnormalities [4,9,11]; some of these cases could be regarded as a form of caudal duplication [2,6,14] and in some cases, there is a cleft in the spine rather than two formed spinal columns with two hemi-cords [3]. To the best of our knowledge, any of the reported cases of duplication of spine describes a lipomyelomeningocele as in our case.…”

Section: Discussionmentioning

confidence: 55%

Duplication of spine with hemi-lipomyelomeningocele

2013

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Background Duplication of the spine is very rare, and this malformation is generally considered as a severe form of type I split cord malformations. To the best of our knowledge, this is the first reported case of spine duplication associated with lipomyelomeningocele. Case We report an exceptional case of 14-year-old, asymptomatic and neurologically intact girl with duplication of the spine and marked separation of bony elements at thoraco-lumbar region. One of the split thecal sacs includes a tethered spinal cord whereas other thecal sac has no visible neural content, and there is a neighbor lipomyelomeningocele located in the midline. Conclusion A surgical operation was planned to release the tethered cord and instrumentation and fusion for scoliosis; however, the operation was declined by the patient.

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Section: Discussionmentioning

confidence: 55%

Duplication of spine with hemi-lipomyelomeningocele

2013

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“…Pang et al 2 advanced a unified theory for the spinal cord duplication disorders, suggesting that all result from abnormal adherence between ectoderm and endoderm 2 . In the view of Dominguez et al these anomalies originate from damage to the mass formed by caudal cells and posterior gut at approximately 25 days of pregnancy 1 . Pang et al classified spinal cord duplication anomalies into types I and II.…”

Section: Discussionmentioning

confidence: 99%

“…The more serious forms are rare and only a limited number of cases are on record 1 . They are usually associated with other systemic malformations, including duplication of vascular structures, of the distal gastrointestinal and urogenital tracts (as in the present case), and possibly limb malformations.…”

Section: Discussionmentioning

confidence: 99%

Caudal duplication syndrome

Taneja

Zaffani²,

Amato-Filho

et al. 2009

Arq. Neuro-Psiquiatr.

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“…Caudal duplication syndrome (CDS) as described Domiguez et al in 1993 is a rare association between malformations and duplications of the gastrointestinal and genitourinary systems and the neural tube defects [1]. Although less than 100 cases have been reported in literature worldwide since 1953, a large variation in presentation have been noted.…”

Section: Introductionmentioning

confidence: 99%

“…It is a rare congenital malformation with frequency not well known. The pathophysiology is complex and aetiology unknown and majority of them being diagnosed in neonatal or in paediatric age group [1]. One adult female was reported to have diagnosed with CDS while evaluation for her infertility but she too had undergone unknown surgery in childhood for possible anal malformation [2].…”

Section: Introductionmentioning

confidence: 99%

A Rare Presentation of Caudal Duplication Syndrome in an Adult with No Functional Impairment

Deshpande¹,

Goil²,

Bannerjee³

2014

JCR

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Abstract:Caudal duplication syndrome is a rare congenital association between malformations and duplications of the gastrointestinal and genitourinary systems and neural tube defects. Less than 100 cases have been reported in literature worldwide, majority being diagnosed at birth. To date only 2 patients have been reported to have presented in adulthood. We hereby report a case of caudal duplication with complete duplication of urethra, bladder, vagina, uterus, entire hindgut and midgut but with no functional abnormalities. Her only complaint being narrow double vaginal vaults with anticipated difficulty in sexual function. As normal pregnancy has been reported in uterus didelphys, a simple vaginal septal resection and posterior vaginal wall lengthening with incorporation of a 'Z-plasty' was done along with vulvaplasty to exclude the abnormal anal opening from the fourchette.

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Caudal Duplication Syndrome

Cited by 58 publications

References 8 publications

Duplication of spine with hemi-lipomyelomeningocele

Duplication of spine with hemi-lipomyelomeningocele

Caudal duplication syndrome

A Rare Presentation of Caudal Duplication Syndrome in an Adult with No Functional Impairment

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