Split notochord syndrome in an adult presenting with colonic duplication

Kurisu, Akane; Kawachi, Yasuyuki; Maruyama, Hiroshi; Kuwabara, Noriyuki

doi:10.1007/bf02053344

Cited by 5 publications

(4 citation statements)

References 10 publications

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“…13 Above all, it is now known that few vertebral anomalies (especially the longitudinal defect in vertebral bodies and arches) can be missed on chest radiographs, but can be detected by tomography. 11 The presence of thoracic vertebral anomalies in our patient in all likelihood is a part of split notochord syndrome associated with GD. However, in absence of autopsy, we cannot emphatically rule out the simultaneous presence of thoracic duplication cyst, a known association with GD.…”

Section: Discussionmentioning

confidence: 56%

“…The vertebral anomalies have rarely been reported with isolated infradiaphragmatic abdominal duplications 6 or with GD. 7 Kurisu et al 11 have described a colonic duplication with split notochord syndrome. In their opinion, "split notochord" is the most plausible theory for alimentary duplications, and even in those reported cases where description of the spine is not given this syndrome might have been involved.…”

Section: Discussionmentioning

confidence: 99%

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Unusual Presentation of Gastric Duplication Cyst in a Neonate with Pneumoperitoneum and Vertebral Anomalies

Ratan¹,

Ratan²,

Lohan³

et al. 2002

Am J Perinatol

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An unusual case of communicating, tubular gastric duplication (GD) of the greater curvature of the stomach presenting with pneumoperitoneum is described. The pneumoperitoneum resulted due to simultaneous mechanical rupture of stomach and its duplication cyst due to birth trauma and vigorous post delivery resuscitation. No case of this kind has been reported earlier in English literature, though instances of ulcerative perforation of neonatal stomach and GD are known. There was radiographic evidence of multiple thoracic vertebral anomalies, again a rare occurrence with GD cyst. Further, a diagnostic dilemma was faced in this patient as the presence of radiographic sign of "gastric bubble" on plain radiographs in this patient suggested the source of free intraperitoneal air to be extragastric and the diagnosis could be made only at surgery. The authors have reviewed the pertinent literature on neonatal gastric perforation and GD cysts and uphold "split notochord" theory as etiology for GD in this patient.

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Section: Discussionmentioning

confidence: 56%

Section: Discussionmentioning

confidence: 99%

Unusual Presentation of Gastric Duplication Cyst in a Neonate with Pneumoperitoneum and Vertebral Anomalies

Ratan¹,

Ratan²,

Lohan³

et al. 2002

Am J Perinatol

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“…More subtle anomalies, such as spina bifida occulta, may be indicated only by the presence of hypertrichosis, a nevus, angioma, or a sacral dimple. Associated anomalies are diverse, and those present in our case as mentioned by others [6,8,9,[11][12][13][14][15][16][17][18][19][20][21][22] are listed in Table 1. Bentley and Smith stated that malformations of the heart and the great vessels have also been associated with abnormal spinal development, but a ready explanation is not at hand [6].…”

Section: Discussionmentioning

confidence: 85%

A case of split notochord syndrome: a child with a neuroenteric fistula presenting with meningitis

Ramshorst

Lequin

Mancini

et al. 2006

Journal of Pediatric Surgery

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The authors describe a case of split notochord syndrome with a neuroenteric fistula in a newborn presenting with meningitis. Associated anomalies included agenesis of the corpus callosum, short colon, malrotation, epispadias, and an abnormally high bifurcation of the abdominal aorta and inferior vena cava. The embryological mechanisms and etiologic theories are discussed in short. D 2006 Elsevier Inc. All rights reserved.Split notochord syndrome (SNS) describes congenital spinal anomalies, associated with anomalies of the vertebrae, central nervous system, and gastrointestinal tract. The anomalies arise from a persistent connection between endoderm and dorsal ectoderm. Depending on its size, site, and point in time, abnormal splitting of the notochord can result in a wide variety of malformations and clinical symptomatology. Case reportA 4-day-old boy was referred to our hospital on suspicion of meningitis with secondary dehydration and for evaluation of vertebral anomalies. He was born at 37 weeks of gestation through normal vaginal delivery as the third child of a 26-year-old mother. Birth weight was 3010 g. Physical examination revealed normal tonus, minor facial dysmorphic signs (eg, broad mouth and dysmorphic, normally placed ears with a thin helix), left-sided congenital hip dysplasia, sacral dimple, and rocker bottom feet. The penis showed a mild epispadias, whereas anus, scrotum, and testes were normal. The mother had smoked during pregnancy and had not used supplementary folic acid. Besides local treatment of a vaginal yeast infection, no other medication had been used during pregnancy. The day before, the patient had been brought in at the referring hospital with a 2-day history of poor feeding and fever. He was jaundiced and showed a tachycardia at a body temperature of 398C. Neurologic examination revealed high tonus and a bulging anterior fontanel. The abdomen was distended. Abdominal x-ray showed a spinal cleft of the vertebrae of Th 12 and L1 (Fig. 1). The kidneys were normal on ultrasound. Blood tests showed elevated C-reactive protein (109 mg/L) and white blood cell count (25,500 cells per cubic millimeter). No lumbar puncture was attempted because of the vertebral anomalies. Blood cultures showed growth of Escherichia coli bacteria, after which, antibiotic treatment was switched from amoxicillin/ cefotaxim to meropenem.

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“…The cases involving the lumbar region are very rare. There has been a case report of tubular colonic duplication, sigmoid colon diverticulum, and lumbar anterior spina bifida in a 28-year-old man diagnosed with split notochord syndrome (Kurisu et al 1992). The patient was exceptional in having reached the age of 28 years without complaints.…”

Section: Discussionmentioning

confidence: 97%

Split Notochord Syndrome: Ileal Duplication Causing Intermittent Episodes of Vomiting

Tsuchida

Takahashi

Noguchi

et al. 2006

Tohoku J. Exp. Med.

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Split notochord syndrome in an adult presenting with colonic duplication

Cited by 5 publications

References 10 publications

Unusual Presentation of Gastric Duplication Cyst in a Neonate with Pneumoperitoneum and Vertebral Anomalies

Unusual Presentation of Gastric Duplication Cyst in a Neonate with Pneumoperitoneum and Vertebral Anomalies

A case of split notochord syndrome: a child with a neuroenteric fistula presenting with meningitis

Split Notochord Syndrome: Ileal Duplication Causing Intermittent Episodes of Vomiting

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