Multidisciplinary Evaluation of Occult Spinal Dysraphism in 47 Children

Sarıca, Kemal; Erbağcı, Ahmet; Yağcı, Faruk; Yurtseven, Cihanser; Buyukbebeci, Orhan; Karakurum, Gunhan

doi:10.1080/00365590310004725

Cited by 23 publications

(14 citation statements)

References 13 publications

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“…Current case series is from countries with relatively high prevalence of BD (420 per 100,000 population in Turkey; 80 per 100,000 population in Iran) [16][17][18]. On the other hand, ALS is relatively rare in these countries (1.57 per 100,000 population in Iran [19]).…”

Section: Discussionmentioning

confidence: 99%

Association of amyotrophic lateral sclerosis and Behcet’s disease: is there a relationship? A multi-national case series

et al. 2012

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Neurological involvement may be seen in 5-30% of the patients with Behcet's disease (BD). Occasionally, parenchymal neurological involvement in BD can present as a spinal cord syndrome. However, motor neuron disease-like presentation is extremely uncommon. Here we are reporting five patients (all male; median age, 38) fulfilling both International Study Group criteria for BD and El Escorial criteria for amyotrophic lateral sclerosis (ALS). These patients were identified by a questionnaire sent to the members of the Neuro-Behcet Study Group of the International Study Group for BD. Three out of five patients had only motor presentations. In two patients, sensory and urinary manifestations were present as well. Spinal cord MRIs were normal in all, and brain MRIs were normal in four patients; one patient had nonspecific white matter changes. Two patients passed away 1-3 years after diagnosis of ALS, and two patients were lost to follow-up 3 and 11 years after admission; one patient is still alive 3 years after onset. The patients that are presented here might represent a rare form of neurological involvement in BD as well as sole coincidence. Larger prospective series are needed to further answer this issue.

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Section: Discussionmentioning

confidence: 99%

Association of amyotrophic lateral sclerosis and Behcet’s disease: is there a relationship? A multi-national case series

et al. 2012

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“…Regular follow-up by specialized nurse practitioners, and enhancing motivation in the parents and children were also influential factors. 21 Despite good clinical effects, some children and parents using transanal irrigation found this method very time consuming and too complicated for self-management. To improve this discomfort, it may be important to start a bowel management program initially and provide sufficient time and support to help children with neurogenic bowel disturbance to be independent at the toilet.…”

Section: Discussionmentioning

confidence: 99%

The effects of transanal irrigation as a stepwise bowel management program on the quality of life of children with spina bifida and their caregivers

Choi

Shin

et al. 2013

Spinal Cord

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Study design: Experimental, prospective study. Objectives: Fecal incontinence and constipation affect the quality of life (QOL) of children with spina bifida and their caregivers. We evaluated the clinical efficacy of a stepwise bowel management program on QOL for children with spina bifida and their caregivers. Setting: Republic of Korea. Methods: Between December 2010 and April 2011, 53 children with constipation, fecal incontinence or both underwent a stepwise bowel management program at our spina bifida clinic. The children and their caregivers were evaluated before and after this program using a self-administered questionnaire. Results: Among the children, 11.3% received only oral laxatives and controlled well, 88.7% received transanal irrigation. After this program, the mean number of episodes of fecal incontinence per week, number of diaper changes and total time for bowel care decreased from 6.9 to 0.5 (P ¼ 0.004), from 1.6 to 0.2 (P ¼ 0.001) and from 27 to 15.9 min (P ¼ 0.003), respectively. Caregivers and children were able to leave their houses more often (P ¼ 0.006), and caregivers' bothersomeness, anxiety and depression due to bowel care decreased (Po0.001). Factors related to family relationships (P ¼ 0.265) and financial impact (P ¼ 0.071) improved, but not significantly. Conclusions: We observed significant improvement in defecation symptoms and QOL scores of spina bifida patients who underwent this program. We recommend that this simple therapeutic method be considered as a safe and valid choice for the treatment of chronic constipation and fecal incontinence.

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“…Other important factors that could have affected this result were the financial, social, economic, educational and intellectual level of the newborns’ families. The rate of spinal-dysraphism-related nervous system abnormalities in neonates with a single and small lesions is low [26]; however, the study of 171 neonates who had spinal dysraphism abnormalities showed that multiple skin lesions, large subcutaneous masses and cutaneous sinuses over the sacrum were correlated with an increase in the rate of spinal dysraphism disorders. Henriques et al [10] also prospectively studied minor skin lesions as signs of spinal dysraphism by spinal USG.…”

Section: Discussionmentioning

confidence: 99%

Correlation of Cutaneous Lesions with Clinical Radiological and Urodynamic Findings in the Prognosis of Underlying Spinal Dysraphism Disorders

Sasani

Asghari²,

Asghari

et al. 2008

Pediatr Neurosurg

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Spinal dysraphism is characterized by a lack of fusion of the vertebral arches that occurs in the absence of spinous processes with variable amounts of lamina. Here, we retrospectively present the importance of cutaneous lesions and their correlation with clinical presentation, radiological examination and urodynamic assessment. We retrospectively reviewed 612 (6.12%) cases with skin lesions from 10,000 consecutive live-born children seen at two institutions between January 1998 and March 2005. We divided all children into a control group and three groups based on clinical assessment, radiological examination and urodynamic evaluation results. Neurological deficits were identified in 113 (18.46%) children, while spinal dysraphism disorders, tethered cord syndrome and associated orthopedic malformations were found in 171 (27.94%), 119 (19.45%) and 28 (4.57%) patients, respectively. The incidence of tethered cord with lumbosacral dimple lesions over the sacrum (32/119, 26.65% of patients) was 3.5-fold higher than that of lesions found over the coccyx (9/119, 7.5% of patients). Uroneurological symptoms were found in 207/612 (33.82%) children. Urodynamic assessment revealed decreased bladder capacity in 10% of patients, detrusor hyperflexia during filling in 47% and a low-compliance detrusor in 71%. Discordance between ultra- sonography and MRI was found in 16.58% of patients. Spinal cord untethering was performed for 109 patients. Nearly all children with resolution were at the end of their follow-up period (24 months). Retethering occurred in 21 (19.26%) patients, and a second untethering surgery was performed in 12 patients. When spina bifida was associated with lumbar skin lesions, there may have been an increased incidence of tethered cord and other spinal cord disorders. MRI scans are more reliable and give an exact diagnosis of tethered cord. Neurological and uroneurological instability are ultimately a clinical diagnosis, and there is controversy about their indications for surgery. However, the correlation between urodynamic assessment and cutaneous lesions with a tethered cord found by MRI examination allow for an early diagnosis and the possibility of prompt treatment.

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Multidisciplinary Evaluation of Occult Spinal Dysraphism in 47 Children

Cited by 23 publications

References 13 publications

Association of amyotrophic lateral sclerosis and Behcet’s disease: is there a relationship? A multi-national case series

Association of amyotrophic lateral sclerosis and Behcet’s disease: is there a relationship? A multi-national case series

The effects of transanal irrigation as a stepwise bowel management program on the quality of life of children with spina bifida and their caregivers

Correlation of Cutaneous Lesions with Clinical Radiological and Urodynamic Findings in the Prognosis of Underlying Spinal Dysraphism Disorders

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