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Martin M. Tisdall, Richard D. Hayward, and Dominic N. P. Thompson

OBJECT

A dermal sinus tract is a common form of occult spinal dysraphism. The presumed etiology relates to a focal failure of disjunction resulting in a persistent adhesion between the neural and cutaneous ectoderm. Clinical and radiological features can appear innocuous, leading to delayed diagnosis and failure to appreciate the implications or extent of the abnormality. If it is left untreated, complications can include meningitis, spinal abscess, and inclusion cyst formation. The authors present their experience in 74 pediatric cases of spinal dermal tract in an attempt to identify which clinical and radiological factors are associated with an infective presentation and to assess the reliability of MRI in evaluating this entity.

METHODS

Consecutive cases of spinal dermal tract treated with resection between 1998 and 2010 were identified from the departmental surgical database. Demographics, clinical history, and radiological and operative findings were collected from the patient records. The presence or absence of active infection (abscess, meningitis) at the time of neurosurgical presentation and any history of local sinus discharge or infection was assessed. Magnetic resonance images were reviewed to evaluate the extent of the sinus tract and determine the presence of an inclusion cyst. Radiological and operative findings were compared.

RESULTS

The surgical course was uncomplicated in 90% of 74 cases eligible for analysis. Magnetic resonance imaging underreported the presence of both an intradural tract (MRI 46%, operative finding 86%) and an intraspinal inclusion cyst (MRI 15%, operative finding 24%). A history of sinus discharge (OR 12.8, p = 0.0003) and the intraoperative identification of intraspinal inclusion cysts (OR 5.6, p = 0.023) were associated with an infective presentation. There was no significant association between the presence of an intradural tract discovered at surgery and an infective presentation.

CONCLUSIONS

Surgery for the treatment of spinal dermal tract carries a low morbidity. While it seems intuitive that tracts without intradural extension carry a low risk of spinal cord tethering, it is not possible to reliably detect these cases using MRI. Similarly, intraspinal dermoid cannot be reliably excluded using MRI and carries an increased risk of infection. These points justify excision together with intradural exploration of all spinal dermal sinus tracts.

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Olivier Klein and Dominic Thompson

✓The authors present the case of a 3-month-old girl who presented with lipomyelomeningocele (LMM) associated with a low-lying conus and a terminal syrinx. At the time of presentation, the girl had a mild neurological, but congenital, deficit: weakness of the right foot and calf as well as absence of the right ankle reflex. Bladder assessment was normal, and it was decided to monitor this child closely, without performing surgery. Results of further magnetic resonance imaging performed 1 year after diagnosis and when the girl was 28 months old, showed a significant regression of LMM and associated syrinx. Interestingly, the child remained neurologically unchanged. Although described in the literature, spontaneous regression of LMM is a very unusual situation, and very few cases have been reported. This case represents the first report of concomitant regression of an associated syrinx. Close follow-up of nonsurgically treated children is mandatory, as the risk of late deterioration remains.

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Dora Steel, Matthew A. Kirkman, Dominic N. P. Thompson, and Kristian Aquilina

Open anterolateral cordotomy is an effective treatment option for adults with intractable pain, but it has seldom been attempted in children. In the 2 previously reported cases in children, the procedure was used within 10 days of death from neoplastic disease. In this paper the authors describe 2 cases in which open cordotomy was used successfully in children outside the immediate terminal phase of disease. Both patients experienced effective analgesia with minimal adverse effects. The authors propose that consideration of cordotomy as an option for the management of intractable pain in children does not need to be delayed until death is imminent.

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Juan F. Martínez-Lage

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Michael D. Cearns, Samantha Hettige, Paolo De Coppi, and Dominic N. P. Thompson

OBJECTIVE

It is well established that Currarino syndrome (CS) may be associated with spinal dysraphism. Here, the authors report on 10 CS patients with dysraphic anomalies who had undergone a combined neurosurgical and general surgical approach to repair the dysraphic anomalies and resect the presacral mass in a single operation. They discuss the spectrum of spinal dysraphism that may coexist in CS in the context of its developmental etiology.

METHODS

Children with a confirmed CS diagnosis who had undergone the combined operative approach were identified from a departmental database. Presenting features were recorded and preoperative imaging was analyzed to record features of the presacral mass and the dysraphic anomalies. The histopathological nature of the resected presacral mass and the outcomes postoperatively and at the last follow-up were reviewed.

RESULTS

Between 2008 and 2015, 10 patients presented with CS, 9 with constipation. Median age at the time of surgery was 1.3 years. Six of the 10 patients had anorectal malformation consisting of anal stenosis, rectal stenosis, or imperforate anus. Spinal anomalies included anterior meningocele (5 cases), low-lying conus (8), terminal syrinx (4), fatty filum (5), caudal lipoma (3), and intraspinal cyst (1). In all cases, the lumbosacral spinal canal was accessed via a midline approach with laminoplasty, allowing spinal cord untethering and repair of the dysraphic anomalies. Following dural closure, the incision was extended inferiorly to incorporate a posterior sagittal approach to resect the presacral mass. The histopathological nature of the mass was mature teratoma (8 cases), complex hamartomatous malformation (1), or neurenteric cyst (1). There were no new instances of neurological deterioration, with most instances of persisting morbidity related to constipation (6 cases) or neurogenic bladder dysfunction (8). There were no infective complications, no instances of cerebrospinal fluid fistula, no recurrences of the presacral mass, and no cases of retethering of the spinal cord.

CONCLUSIONS

Although not part of the original triad, spinal dysraphic anomalies are common in CS and in keeping with a disorder of secondary neurulation. Lumbosacral MRI is an essential investigation when CS is suspected. Children are at risk of sphincter impairment due to the anorectal malformation; however, both spinal cord tethering and compression from the presacral mass may further compromise long-term continence. A combined operative approach to repair the dysraphic anomalies and resect the presacral mass is described with good postoperative and long-term outcomes.

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Ali Nader-Sepahi, Adrian T. H. Casey, Richard Hayward, H. Alan Crockard, and Dominic Thompson

Object

The aim of this study was to audit the treatment of symptomatic atlantoaxial instability in Down syndrome and to assess factors associated with fusion failure in this group of patients.

Methods

The authors reviewed the cases of 12 children with Down syndrome presenting with symptomatic atlantoaxial instability who underwent surgery for internal fixation and fusion. A review of clinical histories, radiological investigations, and details of operative interventions was also performed.

The mode of presentation was acute spinal cord injury (five cases), progressive myelopathy (four cases), and neck pain or stiffness (three cases). The atlantodental interval ranged from 5 to 13 mm. The posterior atlantodental interval at the C-1 level was 9.5 mm (range 6–11 mm). In 10 patients an os odontoideum was present. Translocation of the odontoid process occurred in one patient, and occipitoatlantal instability was also identified in two cases. Twenty-four operations were performed in the 12 patients. A transoral odontoidectomy was required in four children. Successful fusion was demonstrated in seven patients at the first operation. Three of the five patients with acute cord injury made significant functional recovery and were left with no or mild disability.

Conclusions

Additional bone abnormalities at the CVJ are common in the Down syndrome population. Young age at the time of fusion and multiple osseous anomalies pose a higher risk factor in fusion failure. The authors recommend an aggressive surgical approach for management of symptomatic cases of CVJ instability.

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John E. Crossman, Dominic Thompson, Richard D. Hayward, Andrew O. Ransford, and H. Alan Crockard

✓ Atlantoaxial rotatory fixation (AARF) is an uncommon condition of childhood. Occasionally AARF may recur. The authors describe the cases of four patients with recurrent AARF (RAARF).

The probable cause of the RAARF and operative procedure selected are discussed. In three cases, attempts were made to stabilize the atlantoaxial complex rather than to perform fusion to preserve the function of the joint complex.

Joint stabilization is performed by incorporating a “check ligament” into the joint construct between the axial spinous process and the atlantal lateral mass. The authors believe this technique of joint stabilization augments the strength of the joint, allowing normal, but preventing excessive, rotation, until the joint reaches physiological maturity.

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Charles Kondageski, Dominic Thompson, Mandy Reynolds, and Richard D. Hayward

Object

The overdrainage of cerebrospinal fluid (CSF) in children with shunt-treated hydrocephalus may cause chronic disabling symptoms and require repeated surgery. Externally adjustable valves offer a noninvasive way of altering the valve opening pressure. The authors report on their experience with using the Strata valve in the management of symptomatic CSF overdrainage.

Methods

The authors treated 24 patients with symptomatic CSF overdrainage by inserting a Strata valve. The severity of symptoms was graded, and the frequency of hospital visits and shunt operations was recorded before and after insertion of the valve. Additionally, results of brain imaging and intracranial pressure monitoring were reviewed. Nineteen patients (79.2%) had severe symptoms at the time of the insertion; 1 year after Strata valve insertion only one patient (4.17%) still suffered severe symptoms. The number of hospital admissions was 3.38/patient/year before placement and 1.21 for the 1st year, 1 for the 2nd, and 0.4 for the 3rd postoperative year. The number of operations was 3.42/patient/year during the year before placement of the valve, and then 0.71 for the 1st, 0.56 for the 2nd, and 0.25 for the 3rd postoperative years. During the 1st year after placement of the Strata valve, the settings were changed 2.79 times/patient/year, 1.29 for the 2nd, and 1.33 times/patient/year for the 3rd year.

Conclusions

The Strata valve was effective in improving the symptoms of overdrainage in the majority of patients in this series. The number of hospital admissions and operations for valve malfunction was reduced.