[Effects of hypocaloric diet on respiratory manifestations in Willi-Prader syndrome]

Arch Pediatr. 1995 Nov;2(11):1075-9. doi: 10.1016/0929-693x(96)81283-8.
[Article in French]

Abstract

Background: Prader-Willi syndrome (PWS) is a multisystem disorder with hyperphagia and obesity. Breathing disorders such as snoring, sleep apnea syndrome, and sleep hypoventilation have also been reported.

Case report: Jonathan was born with the typical features of PWS. He developed exercise dyspnea, cyanosis and nocturnal sweating at the age of 3 years. A few months later, a respiratory failure required his admission to the intensive care unit. His weight was then 48 kg (300% ideal body weight (IBW); physical examination showed polypnea (60 breaths/min), cyanosis, fat deposition on the chest wall. Transcutaneous oxygen saturation was 65%, carbon dioxide tension 81 mmHg (capillary sample). Pulmonary hypertension was found (mean arterial pulmonary pressure = 55 mmHg). Polysomnography detected hypoventilation with persistent hypoxemia increasing during the night and transient dips of oxygen saturation with bradycardia. He was treated with oxygen, mechanical ventilation (facial mask) and a low caloric diet (600 cal/day). Four months later, he weighed 33 kg (200% IBW); the respiratory features had resolved and gazometric values and pulmonary pressure returned to the normal ranges. Polysomnography showed only obstruction apnea and hypopnea without oxygen desaturation.

Conclusions: Patients with PWS may develop respiratory symptoms sufficient by severity to be life threatening. They are related to morbid obesity and are influenced by a hypocaloric diet. Follow-up of patients with this syndrome must include repeated respiratory evaluation.

Publication types

  • Case Reports
  • English Abstract

MeSH terms

  • Child, Preschool
  • Diet, Reducing
  • Energy Intake
  • Humans
  • Male
  • Obesity / diet therapy
  • Obesity / etiology
  • Prader-Willi Syndrome / complications*
  • Prader-Willi Syndrome / diagnosis
  • Respiration Disorders / diet therapy*
  • Respiration Disorders / etiology*