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Tomomi Kimiwada, Toshiaki Hayashi, Reizo Shirane, and Teiji Tominaga

OBJECTIVE

Some pediatric patients with moyamoya disease (MMD) present with posterior cerebral artery (PCA) stenosis before and after anterior circulation revascularization surgery and require posterior circulation revascularization surgery. This study evaluated the factors associated with PCA stenosis and assessed the efficacy of posterior circulation revascularization surgery, including occipital artery (OA)–PCA bypass, in pediatric patients with MMD.

METHODS

The presence of PCA stenosis before and after anterior circulation revascularization surgery and its clinical characteristics were investigated in 62 pediatric patients (< 16 years of age) with MMD.

RESULTS

Twenty-three pediatric patients (37%) with MMD presented with PCA stenosis at the time of the initial diagnosis. A strong correlation between the presence of infarction and PCA stenosis before anterior revascularization was observed (p < 0.001). In addition, progressive PCA stenosis was observed in 12 patients (19.4%) after anterior revascularization. The presence of infarction and a younger age at the time of initial diagnosis were risk factors for progressive PCA stenosis after anterior revascularization (p < 0.001 and p = 0.002, respectively). Posterior circulation revascularization surgery, including OA-PCA bypass, was performed in 9 of the 12 patients with progressive PCA stenosis, all of whom showed symptomatic and/or radiological improvement.

CONCLUSIONS

PCA stenosis is an important clinical factor related to poor prognosis in pediatric MMD. One should be aware of the possibility of progressive PCA stenosis during the postoperative follow-up period and consider performing posterior circulation revascularization surgery.

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Toshiaki Hayashi, Tomomi Kimiwada, Reizo Shirane, and Teiji Tominaga

OBJECTIVE

Lipoma of the conus medullaris (LCM) causes neurological symptoms known as tethered cord syndrome (TCS). The symptoms can be seen at diagnosis and during long-term follow-up. In this report, pediatric patients with LCMs who underwent untethering surgery, under the policy of performing surgery if diagnosed regardless of symptoms, were retrospectively reviewed to evaluate long-term surgical outcomes. Possible risk factors for retethered cord syndrome (ReTCS) were evaluated in the long-term follow-up period.

METHODS

A total of 51 consecutive pediatric patients with LCMs who underwent a first untethering surgery and were followed for > 100 months were retrospectively analyzed. The surgery was performed with the partial removal technique. Pre- and postoperative clinical and radiological data were reviewed to analyze the outcomes of surgery and identify potential risk factors for ReTCS.

RESULTS

During follow-up, 12 patients experienced neurological deterioration due to ReTCS. The overall 10-year and 15-year progression-free survival rates were 82.3% and 75.1%, respectively. On univariate analysis, a lipoma type of lipomyelomeningocele (OR 11, 95% CI 2.50–48.4; p = 0.0014), patient age at the time of surgery (OR 0.41, 95% CI 0.14–1.18; p = 0.0070), and the mean patient growth rate after surgery (OR 2.00, 95% CI 1.12–3.41; p = 0.0040) were significant factors associated with ReTCS. Cox proportional hazard models showed that a lipoma type of lipomyelomeningocele (HR 5.16, 95% CI 1.54–20.1; p = 0.010) and the mean growth rate after surgery (HR 1.88, 95% CI 1.00–3.50; p = 0.040) were significantly associated with the occurrence of ReTCS.

CONCLUSIONS

More complex lesions and a high patient growth rate after surgery seemed to indicate increased risk of ReTCS. Larger prospective studies and registries are needed to define the risks of ReTCS more adequately.

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Tomomi Kimiwada, Toshiaki Hayashi, Ayumi Narisawa, Reizo Shirane, and Teiji Tominaga

OBJECT

Some pediatric patients with middle cranial fossa arachnoid cysts present with symptoms of increased intracranial pressure (ICP) and require shunt placement after a cyst fenestration. However, factors concerning increased ICP after fenestration followed by shunt placement have not been elucidated. This study evaluated factors that are associated with shunt placement following cyst fenestration in pediatric patients with middle cranial fossa arachnoid cysts.

METHODS

Twenty-six pediatric patients with middle cranial fossa arachnoid cysts who were surgically treated at a single institution between 2004 and 2013 were retrospectively identified. The surgical indications for middle cranial fossa arachnoid cysts were as follows: 1) arachnoid cysts associated with symptoms such as headache and abnormally enlarging head circumference; 2) progressively expanding arachnoid cysts; and 3) large arachnoid cysts such as Galassi Type III. A cyst fenestration was performed as a first-line treatment, and shunt placement was required if symptoms associated with increased ICP were found following fenestration. The risk factors evaluated included age, sex, presenting symptoms, the presence of head enlargement, progressive cyst expansion, and subdural hematoma/hygroma.

RESULTS

Four patients (15.4%) required shunt placement after cyst fenestration. Younger age, abnormal head enlargement, and progressive cyst expansion before fenestration were significantly associated with the need for shunt placement following fenestration. Arachnoid cysts decreased in size in 22 patients (84.6%) after fenestration and/or shunt placement. The presence of symptoms was not associated with postoperative cyst size in this study.

CONCLUSIONS

In this study, younger age, abnormal head enlargement, and progressive cyst expansion were risk factors for shunt placement after cyst fenestration in pediatric patients with middle cranial fossa arachnoid cysts. It is important to consider that cyst fenestration may not be effective because of a latent derangement of CSF circulation in patients with these risk factors.

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Toshiaki Hayashi, Reizo Shirane, Michiko Yokosawa, Tomomi Kimiwada, and Teiji Tominaga

Object

The rate of infection following shunt procedures is unacceptably high. The authors have hypothesized that the key to reducing the shunt infection rate is in reducing bacteria in the operating field and wound. This hypothesis has been tested in a prospective nonrandomized controlled manner.

Methods

Data obtained in all patients undergoing shunt insertions or revisions for hydrocephalus performed between October 1, 2003, and June 12, 2009, were reviewed. Starting in August 2006, we began routinely irrigating the operating field and wound with saline solution from a syringe. Prior to this, we had not used any irrigation techniques, providing an adequate control group (Group A) for the effect of the irrigation technique. Prior to November 2007, we used saline containing amikacin for irrigation (Group B). After that date, we used saline only for irrigation (Group C).

Results

A total of 150 shunt procedures were performed in 79 girls and 71 boys during the study period. The mean age of all patients was 44.0 ± 59.1 months. Groups A, B, and C comprised 61, 40, and 49 shunt procedures, respectively. There was no statistical difference in age among the 3 groups. Nine infections occurred within 90 days in the postoperative period. The overall infection rate was 6.0%. Eight infections occurred before introducing the irrigation procedure (infection rate 13.1%). One infection was noted after introducing irrigation (Group B [0.0%] + Group C [2.0%]; combined B and C infection rate = 1.1%). There was a statistical difference in the infection rate between Group A and Groups B and C combined (p = 0.003), Groups A and B (p = 0.021), and Groups A and C (p = 0.035). In contrast, no statistical difference was observed between Groups B and C (p > 0.99). Six of the 9 infections were due to staphylococcal species.

Conclusions

An irrigation technique used to reduce bacteria in the operating field and wound is effective for preventing shunt infection. Irrigation alone, and not antibiotics, contributed to the prophylaxis of shunt infection.

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Toshiaki Hayashi, Jun Takemoto, Tatsuhiro Ochiai, Tomomi Kimiwada, Reizo Shirane, Kiyohide Sakai, Haruo Nakagawa, and Teiji Tominaga

Object

After untethering of spinal dysraphism, some patients present with neurological deterioration, defined as retethered cord syndrome. It is known that surgical untethering is an option for improving the symptoms of retethered cord syndrome. Previous reports have shown that postoperative improvement in retethered cord syndrome was noted in the majority of patients presenting with pain, and in more patients with motor weakness than in those with urological symptoms. The authors speculate that subjective symptoms may be detected while symptoms are still reversible. In contrast, changes in urological function are less easy to diagnose, and delays in treatment may be complicated by advanced symptoms. In this study, patients with retethered cord syndrome were evaluated to investigate the benefits of performing routine urodynamic study to detect detrusor overactivity, which is considered to be a subclinical change of urological function, and to investigate the efficacy of early untethering surgery on the symptoms of retethered cord syndrome.

Methods

Surgical indications and outcomes of 78 untethering operations (20 for myelomeningocele, 58 for spinal lipoma) for retethered cord syndrome were examined. Diagnosis of retethered cord syndrome was defined by a multidisciplinary spina bifida team, and included routine urodynamic study.

Results

Preoperative symptoms included urological symptoms (70%), lower-extremity symptoms (45%), and others. The most frequent urological symptom was detrusor overactivity detected by urodynamic study (88.7%). Urinary incontinence was only found in 9.4% of patients. Postoperatively, progressive motor weakness improved in all patients, and sensory symptoms improved in 94%. Urological symptoms improved in 80% of the patients with urinary incontinence and in 75% of the patients with detrusor overactivity. Postoperative urodynamic study showed a significant increase in bladder volume (p < 0.05). The most common complication was temporary lower leg paresthesia that recovered at follow-up. Aggravated dysuria was noted in 3 patients.

Conclusions

Early untethering operations offer symptomatic relief to patients with retethered cord syndrome. Urodynamic study findings, especially detrusor overactivity, are considered to be the most significant indicators for early diagnosis of retethered cord syndrome.