Noguchi Tadahide, Sugiyama Tomoko, Sasaguri Ken-Ichi, Ono Shigeru, Maeda Kosaku, Nishino Hiroshi, Jinbu Yoshinori, Mori Yoshiyuki
*Department of Dentistry, Oral and Maxillofacial Surgery, Jichi Medical University †Department of Pediatric Surgery, Jichi Medical University, Shimotsuke-shi ‡Department of Pediatric Surgery, Kobe Children's Hospital, Kobe §Department of Otolaryngology, Head and Neck Surgery, Jichi Medical University, Shimotsuke-shi, Japan.
J Craniofac Surg. 2017 Mar;28(2):e141-e144. doi: 10.1097/SCS.0000000000003324.
A 1-day-old male infant was referred to our department for evaluation of multiple malformations in his oral cavity. He was diagnosed duplication of the pituitary gland-plus syndrome with epignathus, cleft palate, duplication of the mandible, and a lobulated tongue. A thumb-sized mass lesion was visible on the hard palate. The duplicated mandible and lower lip was fused at the midline. The alveolar ridge was protruding through a wide-cleft soft palate involving the uvula. Further examination showed a lobulated tongue, which was seen behind the duplicated part of the mandible. Five days after birth, tracheotomy and epignathus resection were performed. At 7 months of age, the excess tissue of the duplicated mandible was resected at the area of adhesion on the lingual side, and the duplicated tongue and lip were reconstructed. A palatoplasty was performed at 20 months of age. Thereafter, the patient's progress was uneventful, with no abnormality in swallowing. No recurrence of epignathus has been observed during 2 years of follow-up.
一名1日龄男婴因口腔多处畸形被转诊至我科。他被诊断为垂体重复综合征合并上颌寄生胎、腭裂、下颌重复及分叶状舌。硬腭可见一个拇指大小的肿块病变。重复的下颌和下唇在中线处融合。牙槽嵴通过累及悬雍垂的宽大软腭裂突出。进一步检查发现分叶状舌,位于重复下颌部分的后方。出生后5天,进行了气管切开术和上颌寄生胎切除术。7个月大时,在舌侧粘连部位切除了重复下颌的多余组织,并对重复的舌和唇进行了重建。20个月大时进行了腭裂修复术。此后,患者病情平稳,吞咽无异常。随访2年未观察到上颌寄生胎复发。