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Nicholas Sader, Walter Hader, Aaron Hockley, Valerie Kirk, Adetayo Adeleye, and Jay Riva-Cambrin

OBJECTIVE

Chiari 1.5 malformation is a subgroup of the Chiari malformation in which tonsillar descent into the foramen magnum is accompanied by brainstem descent. No data exist on whether operative decompression in patients with Chiari 1.5 improves sleep-related breathing disorders (SRBDs) and whether there are radiological parameters predicting improvement.

METHODS

The authors performed a retrospective cohort study of consecutive pediatric patients with Chiari 1.5 malformation and SRBDs at the Alberta Children’s Hospital. An SRBD was characterized using nocturnal polysomnography (PSG), specifically with the apnea-hypopnea index (AHI), the obstructive apnea index, and the central apnea index. Preoperative values for each of these indices were compared to those following surgical decompression. The authors also compared preoperative radiographic factors as predictors to both preoperative AHI and the change in AHI with surgery. Radiological factors included tonsillar and obex descent beneath the basion–opisthion line, the presence of syringomyelia, the frontooccipital horn ratio, the pB–C2 line, and the clivoaxial angle.

RESULTS

Seven patients (5 males, 2 females) met inclusion criteria. One patient had two surgical decompressions, each with pre- and postoperative PSG studies (n = 8). The median age was 9 years. Before surgical decompression, 75% underwent tonsillectomy/adenoidectomy. The majority (87.5%) experienced snoring/witnessed apnea preoperatively. The median tonsillar and obex descent values were 21.3 mm and 11.2 mm, respectively. The median values for the pB–C2 line and clivoaxial angle were 5.4 mm (interquartile range [IQR] 4.5 mm, 6.8 mm) and 144° (IQR 139°, 167°), respectively. There was a statistically significant change from preoperative to postoperative AHI (19.7 vs 5.1, p = 0.015) and obstructive apnea index (4.5 vs 1.0, p = 0.01). There was no significant change in the central apnea index with surgery (0.9 vs 0.3, p = 0.12). No radiological factors were statistically significant in predicting preoperative AHI and change in AHI.

CONCLUSIONS

This is the first series of pediatric patients with Chiari 1.5 with SRBDs who demonstrated a marked improvement in their PSG results postdecompression. Sleep apnea has a significant impact on learning and development in children, highlighting the urgency to recognize Chiari 1.5 as a more severe form of the Chiari I malformation.

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Aaron Hockley, Michael K. Tso, Mohammed A. Almekhlafi, Abhay K. Lodha, Robin Clegg, Jeremy Luntley, Muneer Eesa, and John H. Wong

OBJECTIVE

Vein of Galen aneurysmal malformations (VGAMs) in infancy have a poor natural history if left untreated. Their high-flow nature can preclude safe and accurate therapeutic vessel occlusion and the risk of inadvertent pulmonary embolism is predominant. The authors describe the technique of rapid cardiac ventricular pacing for inducing transient hypotension to facilitate the controlled embolization of VGAMs.

METHODS

Initial transjugular venous access was obtained for placing temporary pacing leads for rapid cardiac ventricular pacing immediately prior to embolization. Definitive transarterial embolization procedures for the VGAMs were then performed in the same setting in which liquid embolic agents or coils were used.

RESULTS

Beginning in 2010, a total of five procedures were performed in three infants. Transvenous rapid cardiac ventricular pacing was successfully achieved to induce systemic transient flow arrest in all but two attempts, and facilitated partial embolization with n-butyl cyanoacrylate (n-BCA) and coils in all procedures. Ventricular fibrillation occurred twice in one patient and was successfully reversed with defibrillation on both occasions. One patient failed to improve and died from refractory heart failure. Two patients stabilized following staged embolization.

CONCLUSIONS

Rapid transvenous cardiac ventricular pacing can be considered to induce transient hypotension and facilitate controlled embolization in challenging high-flow VGAMs.