Cyanotic breath-holding spell: a life-threatening complication after radical resection of a cervicomedullary ganglioglioma.

Abstract:

:Cyanotic breath-holding spell is a benign and self-limiting disease of young children but occasionally associated with sudden, unexpected death. The authors report a rare case in a 2-year-old girl with a severe form that started after radical resection of a cervicomedullary ganglioglioma. She was admitted to our hospital because of delayed and unstable gait. Since magnetic resonance imaging showed a cervicomedullary tumor, she underwent a radical resection and histology showed the tumor to be a ganglioglioma. Postoperatively, the function of the lower cranial nerves and cerebellum deteriorated and hemiparesis on the left became apparent, but she returned to the preoperative state in a few months. In addition, mild sleep apnea (Ondine curse) and severe cyanotic breath-holding spells occurred. The former responded to medication but the latter failed and continued several times per day with a rapid onset and progression of hypoxemia, loss of consciousness, sweating and opisthotonos. Five months after the operation, the patient returned home with a portable oxygen saturation monitor equipped with an alarm. This case indicates that cyanotic breath-holding spell, as well as sleep apnea, is critical during the early postoperative period. This is the first report observing that such spells may occur as a complication of radical resection of a cervicomedullary tumor.

journal_name

Pediatr Neurosurg

journal_title

Pediatric neurosurgery

authors

Fujisawa H,Yoshida Y,Niida Y,Hasegawa M,Yamashita J

doi

10.1159/000085163

keywords:

subject

Has Abstract

pub_date

2005-03-01 00:00:00

pages

93-7

issue

2

eissn

1016-2291

issn

1423-0305

pii

85163

journal_volume

41

pub_type

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