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Outcomes of adenotonsillectomy in patients with Prader-Willi syndrome
Insights
Adenotonsillectomy (T&A) improved sleep-disordered breathing in most Prader-Willi syndrome (PWS) patients with mild to moderate obstructive sleep apnea (OSA). However, T&A may not fully resolve severe OSA and can increase central apneas, necessitating post-operative polysomnography (PSG).
Area of Science:
- Pediatric Surgery
- Sleep Medicine
- Genetics
Background:
- Prader-Willi syndrome (PWS) is associated with an increased risk of sudden death, particularly in children treated with growth hormone.
- Sleep-disordered breathing (SDB) is common in PWS and requires careful monitoring.
- Polysomnography (PSG) is recommended for detecting SDB in PWS patients.
Purpose of the Study:
- To evaluate the effectiveness of upper airway surgical intervention, specifically adenotonsillectomy (T&A), in managing sleep-disordered breathing in pediatric patients with Prader-Willi syndrome.
- To assess changes in polysomnography (PSG) findings before and after T&A in this population.
Main Methods:
- Retrospective study conducted at a multidisciplinary PWS Center.
- Included 13 pediatric patients with PWS who underwent T&A.
- Compared pre-operative and post-operative PSG results to assess SDB resolution.
Main Results:
- Nine of 13 patients (69%) had mild to moderate obstructive sleep apnea (OSA) or obstructive hypoventilation; T&A normalized breathing in 8 of these.
- Four patients (31%) had severe OSA pre-operatively; T&A resolved OSA in 2, but 2 experienced residual obstructive and central apneas post-surgery.
- Genetic analysis showed 61% deletion and 39% uniparental disomy.
Conclusions:
- Adenotonsillectomy is effective for mild to moderate OSA in PWS patients.
- T&A may not be curative for severe OSA in PWS.
- Post-operative PSG is crucial due to potential increases in central apneas after T&A in PWS patients.
Objective:
To assess the efficacy of upper airway surgical intervention in patients with Prader-Willi syndrome (PWS). Due to reports of sudden death in children undergoing treatment with growth hormone for PWS, detection of sleep-disordered breathing by polysomnography (PSG) has been recommended.
Design:
Retrospective study.
Setting:
Multidisciplinary PWS Center at a tertiary care children's hospital.
Patients:
Thirteen pediatric patients with PWS who underwent adenotonsillectomy (T&A) with pre-PSG and post-PSG.
Main Outcome Measures:
Comparison of PSG results before and after T&A.
Results:
Six of our patients were girls (46%); 8 had genetic characteristics consistent with deletion (61%), and the remaining 5 had genetic characteristics consistent with uniparental disomy (39%). The median age at T&A was 3 years (age range, 6 months to 11 years), and the median age at start of growth hormone treatment was 8.5 months (range, 2 months to 6 years). Nine of the 13 patients had mild to moderate obstructive sleep apnea (OSA) or obstructive hypoventilation (69%); in 8 of these 9, breathing normalized after T&A. Four children had severe OSA prior to surgery (31%). Breathing normalized in 2 of these after surgery, but 2 had PSG findings of residual combined obstructive and central apneas postoperatively.
Conclusions:
Adenotonsillectomy, while effective in most children with PWS who demonstrate mild to moderate OSA, may not be curative in children with severe OSA. An increase in central apneas can occur in some children with PWS postoperatively, and it is important to repeat PSG after surgery. Further studies are necessary to determine optimal treatment for some children with PWS and sleep-disordered breathing.
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