PMID- 15793707
OWN - NLM
STAT- MEDLINE
DCOM- 20060809
LR  - 20131121
IS  - 1531-5037 (Electronic)
IS  - 0022-3468 (Linking)
VI  - 40
IP  - 3
DP  - 2005 Mar
TI  - Assessment of obstructive apnea by using polysomnography and surgical treatment 
      in patients with Beckwith-Wiedemann syndrome.
PG  - E17-9
AB  - PURPOSE: Obstructive apnea is sometimes seen in patients with Beckwith-Wiedemann 
      syndrome. The cause of apnea is not limited to macroglossia, and the surgical 
      indication for obstructive apnea has not yet been established. The authors 
      performed polysomnography for the assessment of apnea. METHOD: Overnight 
      polysomnograms were obtained in 2 patients who developed obstructive apnea after 
      1-stage repair for omphalocele. CASE 1: Apnea index (AI), defined as apneic 
      events per hour, indicated 17.3, and SpO2 below 95% occupied 80% of the total 
      sleep time. Computed tomography and magnetic resonance imaging indicated 
      obstruction of the airway between macroglossia and the hypopharynx. Central 
      tongue resection and the division of the frenulum linguae for associated 
      ankyloglossia were performed 97 days after birth. One month after surgery, apneic 
      events disappeared and SpO2 below 95% occupied only 1% of the total sleep time. 
      CASE 2: Obstructive AI indicated 28.1. Division of the frenulum linguae and 
      anterior glossopexy were performed 55 days after birth. Postoperative 
      polysomnogram indicated a marked reduction of AI. CONCLUSIONS: These results 
      indicated that polysomnography was useful for evaluating obstructive apnea and 
      that advancement of the tongue by division of the frenulum linguae may be 
      recommended for the treatment of obstructive apnea in patients with 
      Beckwith-Wiedemann syndrome.
FAU - Kamata, Shinkichi
AU  - Kamata S
AD  - Department of Pediatric Surgery, Osaka University Graduate School of Medicine, 
      Osaka 565-0871, Japan. kamata@pedsurg.med.osaka-u.ac.jp
FAU - Kamiyama, Masafumi
AU  - Kamiyama M
FAU - Sawai, Toshio
AU  - Sawai T
FAU - Nose, Keisuke
AU  - Nose K
FAU - Usui, Noriaki
AU  - Usui N
FAU - Kawahara, Hisayoshi
AU  - Kawahara H
FAU - Fukuzawa, Masahiro
AU  - Fukuzawa M
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - J Pediatr Surg
JT  - Journal of pediatric surgery
JID - 0052631
RN  - S88TT14065 (Oxygen)
SB  - IM
MH  - Beckwith-Wiedemann Syndrome/*complications
MH  - Diaphragm/abnormalities/surgery
MH  - Female
MH  - Hernia, Umbilical/surgery
MH  - Humans
MH  - Hypopharynx/abnormalities
MH  - Infant, Newborn
MH  - Lingual Frenum/surgery
MH  - Macroglossia/complications/surgery
MH  - Oxygen/blood
MH  - *Polysomnography
MH  - Sleep Apnea, Obstructive/blood/diagnosis/*etiology/physiopathology/surgery
EDAT- 2005/03/29 09:00
MHDA- 2006/08/10 09:00
CRDT- 2005/03/29 09:00
PHST- 2005/03/29 09:00 [pubmed]
PHST- 2006/08/10 09:00 [medline]
PHST- 2005/03/29 09:00 [entrez]
AID - S0022346804007766 [pii]
AID - 10.1016/j.jpedsurg.2004.11.011 [doi]
PST - ppublish
SO  - J Pediatr Surg. 2005 Mar;40(3):E17-9. doi: 10.1016/j.jpedsurg.2004.11.011.