Abstract
BackgroundKagami-Ogata syndrome (KOS) is a rare congenital imprinting disorder. The problems related to the anesthetic management of patients with KOS are respiratory distress and difficult endotracheal intubation.Case presentationA 2-year-old male was scheduled to undergo orchiopexy for bilateral cryptorchidism. Although he had a history of severe respiratory distress immediately after birth, his preoperative respiratory condition was stable. He also had marked tracheal deviation. General anesthesia was induced with nitrous oxide and sevoflurane in oxygen. A laryngeal mask airway (LMA) was inserted following rocuronium administration. Anesthesia was maintained with sevoflurane and simultaneous caudal anesthesia. His postoperative course was uneventful.ConclusionsPatients with KOS should preferably undergo elective surgery only after infancy because their respiratory status is more stable as they grow older. Thorough preoperative evaluation of the respiratory tract is important even in KOS patients with a stable respiratory condition.
Highlights
Kagami-Ogata syndrome (KOS) is a rare congenital imprinting disorder
KOS is characterized by craniofacial dysmorphism and thoracic abnormalities
We present the successful anesthetic management of a child with KOS complicated with tracheal deviation
Summary
The issues related to the anesthetic management of patients with KOS are respiratory distress due to thoracic abnormalities and difficult endotracheal intubation due to craniofacial dysmorphism. Detailed preoperative evaluation of the respiratory tract in KOS patients is necessary even in those with a stable respiratory condition. All authors read and approved the final manuscript. Consent for publication Written informed consent was obtained from the parents of the patient for publication of this case report and any accompanying images. Author details 1Department of Dental Anesthesiology, Field of Oral and Maxillofacial Rehabilitation, Kagoshima University Graduate School of Medical and Dental Sciences, 8-35-1 Sakuragaoka, Kagoshima-shi, Kagoshima 890-8544, Japan. Author details 1Department of Dental Anesthesiology, Field of Oral and Maxillofacial Rehabilitation, Kagoshima University Graduate School of Medical and Dental Sciences, 8-35-1 Sakuragaoka, Kagoshima-shi, Kagoshima 890-8544, Japan. 2Department of Anesthesiology, Osaka Women’s and Children’s Hospital, 840 Murodo-cho, Izumi-shi, Osaka 594-1101, Japan. 3Department of Anesthesiology, Osaka Minami Medical Center, 2-1 Kidohigashi-machi, Kawachinagano-shi, Osaka 586-8521, Japan. 4Department of Anesthesiology, Shiga University of Medical Science, Seta Tsukinowa-cho, Otsu-shi, Shiga 520-2134, Japan
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