Multiple neural tube defects in a child: A rare developmental anomaly

Singh, Neha; Singh, Deepak Kumar; Aga, Pallavi; Singh, Ragini

doi:10.4103/2152-7806.104741

Cited by 12 publications

(8 citation statements)

References 6 publications

(7 reference statements)

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“…The locations are not randomly distributed over the neural tube ( P = 2.0*10 −5 ) but show a preference for the occipital region, vertebrae C6 till T3 and the region between T8 and S1 with a peak at L5. (Richards et al, ; Tekkok, ; Etus et al, ; Singh and Singh, ; Srinivas et al, ; Ahmad and Mahapatra, ; Vashu and Liew, ; Bear et al, ; Carmody et al, ; Dankovcik et al, ; Singh and Singh, ; Singh et al, ; Vogel et al, ; Ahmed et al, ; Herman et al, ; Leykamm et al, ; Mahalik et al, ; Meuli et al, ; Sarici et al, ; van Aalst et al, ; Yang et al, ; De la Calle et al, ; Garg et al, ; Gressot et al, ; Hawasli et al, ; Leoni et al, ; Meadows and Hayes, ; Meunier et al, ; Perez da Rosa et al, ; Anand and Mahmoud, ; Canaz et al, ; Hanaei et al, ; Paul et al, ; Radtke et al, ; Ramdurg et al, ; Sargar et al, ; Wood et al, ; Arishima et al, ; Clark and Davidson, ; Dickman et al, ; Dorum et al, ; Santos et al, ; Sharma et al, ; Zhou and Zheng, ). [Color figure can be viewed at http://wileyonlinelibrary.com]…”

Section: Materials and Methods And Resultsmentioning

confidence: 99%

Single‐site neural tube closure in human embryos revisited

et al. 2017

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Since the multi-site closure theory was first proposed in 1991 as explanation for the preferential localizations of neural tube defects, the closure of the neural tube has been debated. Although the multi-site closure theory is much cited in clinical literature, single-site closure is most apparent in literature concerning embryology. Inspired by Victor Hamburgers (1900-2001) statement that "our real teacher has been and still is the embryo, who is, incidentally, the only teacher who is always right", we decided to critically review both theories of neural tube closure. To verify the theories of closure, we studied serial histological sections of 10 mouse embryos between 8.5 and 9.5 days of gestation and 18 human embryos of the Carnegie collection between Carnegie stage 9 (19-21 days) and 13 (28-32 days). Neural tube closure was histologically defined by the neuroepithelial remodeling of the two adjoining neural fold tips in the midline. We did not observe multiple fusion sites in neither mouse nor human embryos. A meta-analysis of case reports on neural tube defects showed that defects can occur at any level of the neural axis. Our data indicate that the human neural tube fuses at a single site and, therefore, we propose to reinstate the single-site closure theory for neural tube closure. We showed that neural tube defects are not restricted to a specific location, thereby refuting the reasoning underlying the multi-site closure theory. Clin. Anat. 30:988-999, 2017. © 2017 Wiley Periodicals, Inc.

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Section: Materials and Methods And Resultsmentioning

confidence: 99%

Single‐site neural tube closure in human embryos revisited

et al. 2017

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“…Only a few cases of triple neural tube defects have been reported in the literature, mostly with discrete defects having intervening normal segments (Table 1). 4,9,11,12 The associated CM-II and hydrocephalus in these cases could be explained by the unified theory of McLone, which is centered on defective apposition of neuroceles during neurulation, resulting in failure to maintain distension of the primitive ventricular system. 5 This lack of distension alters the osteoinductive effect on endochondral bone formation, leading to a small posterior fossa and maldevelopment of the CSF pathway, causing hydrocephalus.…”

Section: Discussionmentioning

confidence: 99%

“…Nevertheless, the unified theory of Pang and the unified theory of McLone are incongruent with a missing link between gastrulation and neurulation. 5,7 There has been only 1 report (by Singh et al) 9 of triple dysraphism with SCM Type II in association with CM-II, evoking an enigmatic link between gastrulation (unified theory of Pang) and neurulation (unified theory of McLone). However, the discrete nature of the lesions in their case imply multiple embryogenetic defects.…”

mentioning

confidence: 99%

Contiguous triple spinal dysraphism associated with Chiari malformation Type II and hydrocephalus: an embryological conundrum between the unified theory of Pang and the unified theory of McLone

Dhandapani

Srinivasan

2016

PED

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Triple spinal dysraphism is extremely rare. There are published reports of multiple discrete neural tube defects with intervening normal segments that are explained by the multisite closure theory of primary neurulation, having an association with Chiari malformation Type II consistent with the unified theory of McLone. The authors report on a 1-year-old child with contiguous myelomeningocele and lipomyelomeningocele centered on Type I split cord malformation with Chiari malformation Type II and hydrocephalus. This composite anomaly is probably due to select abnormalities of the neurenteric canal during gastrulation, with a contiguous cascading impact on both dysjunction of the neural tube and closure of the neuropore, resulting in a small posterior fossa, probably bringing the unified theory of McLone closer to the unified theory of Pang.

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“…Multiple-site neural tube defects (MNTDs) are extremely rare congenital anomalies that are defined by the simultaneous occurrence of more than one NTD in a single case with normal neural tissue in between, generally representing only <1% of the NTD spectrum. 5 , 6 …”

mentioning

confidence: 99%

“… 7 , 8 Split cord formations, although a relatively common association with meningomyelocele, rarely occur with MNTDs. 6 …”

mentioning

confidence: 99%

Multiple-site neural tube defects complicated by multiple-site split cord malformations and thickened filum terminale: experience at a pediatric neurosurgical teaching hospital in Ethiopia. Illustrative case

Shiferaw

Akililu

Worku

et al. 2022

Journal of Neurosurgery: Case Lessons

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BACKGROUND Multiple-site open neural tube defects (MNTDs) and multiple-site split cord malformations (MSCMs) are extremely rare congenital anomalies that are defined by the simultaneous noncontiguous occurrence of more than one neural tube defect (NTD) and split cord malformation (SCM), respectively, in a single case with normal neural tissue in between. This work shows the cooccurrence of MNTDs and MSCMs, which has never been reported in the literature. OBSERVATIONS A single-stage repair for a 13-day-old female neonate with a preoperative diagnosis of MNTDs (thoracic meningocele and thoracolumbar myelomeningocele) plus an additional intraoperative diagnosis of MSCMs (type 3c) of thoracic and thoracolumbar spine, and thickened filum terminale was done with a favorable smooth postoperative course. LESSONS The use of intraoperative meticulous surgical technique along with preoperative skin stigmata helped for anticipation, detection, and treatment of associated complex spinal MNTDs, especially in resource-limited settings, where preoperative magnetic resonance imaging is not routinely used. Whether to repair the MNTDs as a single- versus multiple-stage procedure is mainly a function of the patient’s tolerance to the duration of anesthesia and the anticipated blood loss for the patient’s age. The overall developmental biology and long-term clinical outcome of MNTDs compared to single NTD/SCM is poorly understood and needs further study.

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Multiple neural tube defects in a child: A rare developmental anomaly

Cited by 12 publications

References 6 publications

Single‐site neural tube closure in human embryos revisited

Single‐site neural tube closure in human embryos revisited

Contiguous triple spinal dysraphism associated with Chiari malformation Type II and hydrocephalus: an embryological conundrum between the unified theory of Pang and the unified theory of McLone

Multiple-site neural tube defects complicated by multiple-site split cord malformations and thickened filum terminale: experience at a pediatric neurosurgical teaching hospital in Ethiopia. Illustrative case

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