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Russell-Silver syndrome with cleft palate: a case report
Mami Shiraishi1, Katsuaki Mishima2, Hirotsugu Umeda1
1Department of Oral and Maxillofacial Surgery, Yamaguchi University Graduate School of Medicine, 1-1-1 Minami-kogushi, Ube, Yamaguchi, 755-8505, Japan.
Oral and Maxillofacial Surgery
|December 6, 2018
Summary
This case report details a successful palatoplasty in a young child with Russell-Silver syndrome (RSS) and cleft palate. Careful perioperative management is crucial due to the risk of pharyngeal edema in these patients.
Area of Science:
- Pediatric surgery
- Genetics
- Anesthesiology
Background:
- Russell-Silver syndrome (RSS) presents with intrauterine growth retardation, short stature, and distinct facial features.
- Cleft palate is a rare but significant co-occurrence in RSS, posing unique surgical challenges.
- Previous reports highlight perioperative difficulties like trismus and mandibular hypoplasia in RSS patients with cleft palate.
Observation:
- A female infant with RSS and cleft palate underwent palatoplasty at 3 years and 6 months.
- Despite low weight and limited mouth opening, intubation was manageable, and a mouth gag was fitted.
- Postoperative pharyngeal edema necessitated delayed extubation, with subsequent respiratory stabilization.
Findings:
- Successful surgical correction of cleft palate was achieved in an RSS patient.
- Pharyngeal edema is a notable postoperative complication in RSS patients with cleft palate.
- Perioperative management requires careful consideration of RSS-specific anatomical variations.
Implications:
- This case underscores the importance of anticipating and managing pharyngeal edema in RSS patients undergoing palatoplasty.
- Anesthesiologists and surgeons should be aware of potential airway complications in this population.
- Further research into the specific risks and optimal management strategies for RSS with cleft palate is warranted.
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