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Year : 2007  |  Volume : 2  |  Issue : 1  |  Page : 33-34

Triple meningocele: Cause for a new theory?

Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi, India

Correspondence Address:
Deepak Agrawal
Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi - 110 029
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/1817-1745.32007

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How to cite this article:
Ahmad FU, Agrawal D, Mahapatra A K. Triple meningocele: Cause for a new theory?. J Pediatr Neurosci 2007;2:33-4

How to cite this URL:
Ahmad FU, Agrawal D, Mahapatra A K. Triple meningocele: Cause for a new theory?. J Pediatr Neurosci [serial online] 2007 [cited 2023 Dec 3];2:33-4. Available from: https://www.pediatricneurosciences.com/text.asp?2007/2/1/33/32007

   Case Report Top

A 25-day-old female child presented to us after an uneventful full-term normal vaginal delivery with multiple swellings in the nape of neck and upper back. There was no history of decreased limb movements or cerebrospinal fluid (CSF) leak from any of the swellings. On examination, the child had three swellings in the back of her neck [Figure - 1]. The one in upper neck was 2 cm x 2 cm and was covered with normal healthy skin. Another larger - 5 cm x 6 cm - fluctuant, transilluminant swelling in upper back was covered partially with normal healthy skin and partially with thinned out, atrophic scaly skin. There was another small swelling, around 2.5 cm x 2.5 cm in size, separate from the larger lump above. There was no evidence of inflammation or CSF leak from any of the swellings, and the child had no gross motor or sensory deficits.

Magnetic resonance imaging of the head and spine showed nonvisualization of cerebellum, with a neural mass in the cervico-dorsal region, which herniated posteriorly through a cervico-dorsal defect to form a meningomyelocele, not covered by subcutaneous tissue [Figure - 2]. There was herniation of occipital lobes up to upper cervical level and syringomyelia of dorsolumbar cord. Another small defect in occipital bone caused a small meningocele [Figure - 3].

   Discussion Top

The occurrence of multiple neural tube defects (NTDs) in a single patient is extremely rare. Only nine cases of double NTDs and a single case of triple NTDs have been described in world literature. [1],[2],[3],[4],[5],[6],[7] Almost all these cases have been associated with thoracic and lumbosacral levels. [1],[2],[3],[5],[6] Tekkok [8] presented a rare case of a 2-year-old boy with a parietooccipital encephalocele, a small cervical myelomeningocele and a thoracolumbar myelomeningocele. Hydrocephalus and Chiari II malformation accompanied the NTDs.

The popular theory is that myelomeningocele originates from defective closure of caudal neural tube between 26 and 28 days of gestation. [4] By this theory, however, there can only be two sites of NTDs - the sites of anterior and posterior neuropores. This however does not explain the full range of anomalies seen in humans. [9] VanAllen et al ., in 1993, proposed the "multiple-site closure of neural tube" theory, similar to that observed in mice and other experimental animals, [9] which stated that there are five sites of closure (sites 1 to 5) and NTDs can result from defective closure at any of these sites. Our case too adds credence to this theory, and we believe that multiple factors and sites may be involved in the genesis of neural tube defects.

   References Top

1.Bailey IC. Double meningocele. Arch Dis Child 1971;46:549-50.  Back to cited text no. 1  [PUBMED]  
2.Bertan V, Wilson CB. Double myelomeningocele: A case report. Turk J Pediatr 1968;10:88-90.  Back to cited text no. 2  [PUBMED]  
3.Durmaz R, Arslantas A, Ozon YH, Tel E. Double meningocele: Case report. Turk J Pediatr 2000;42:331-3.  Back to cited text no. 3  [PUBMED]  
4.Hahn YS. Open myelomeningocele. Neurosurg Clin North Am 1995;6:231-41.  Back to cited text no. 4  [PUBMED]  
5.Rainov NG, Heidecke V, Burkert W. Thoracic and lumbar meningocele in neurofibromatosis type 1. Report of two cases and review of the literature. Neurosurg Rev 1995;18:127-34.  Back to cited text no. 5    
6.Richards TA, Kortesis BG, Glazier S, Argenta LC, David LR. Double myelomeningocele: Case report and review. Br J Plast Surg 2003;56:306-8.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Steinbok P. Dysraphic lesions of the cervical spinal cord. Neurosurg Clin North Am 1995;6:367-76.  Back to cited text no. 7  [PUBMED]  
8.Tekkok IH. Triple neural tube defect-cranium bifidum with rostral and caudal spina bifida-live evidence of multi-site closure of the neural tube in humans. Childs Nerv Syst 2005;21:331-5.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Van Allen MI, Kalousek DK, Chernoff GF, Juriloff D, Harris M, McGillivray BC, et al . Evidence for multi-site closure of the neural tube in humans. Am J Med Genet 1993;47:723-43.  Back to cited text no. 9  [PUBMED]  


  [Figure - 1], [Figure - 2], [Figure - 3]

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