A case of congenital unilateral maxillo-mandibular bony fusion in an 8-year-old girl

Koeda, Satoko; Suzuki, Takahiro; Nei, Hitoshi; Inahara, Hanae; Takata, Yoko; Goto, Satoshi; Nagasaka, Hiroshi; Kawamura, Hiroshi

doi:10.1016/j.ajoms.2010.06.003

Cited by 4 publications

(2 citation statements)

References 20 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…3 Most reported cases were neonates with only five cases being older children and adults. 1,2,8,10,11 However in this case mouth opening was decreasing progressively till age of 14 years.…”

Section: Discussionmentioning

confidence: 62%

Unilateral Acquired Posterior Maxillary Mandibular Syngnathia

Sahu

Roy²,

Kashyap³

2016

BJOHNS

View full text Add to dashboard Cite

Introduction: Congenital bony fusion of the maxilla and mandible, especially as an isolated occurrence, is a rare condition while acquired fusion being even rarer. Case report: A rare case of a 32 year old female patient with acquired unilateral bony fusion between the zygomatic arch of maxilla and ramus of mandible is reported. Discussion: Forty one cases of syngnathia has been reported in the international literature from 1936 to 2009, of which 39 cases are congenital and two were acquired, case 1 probably as a result of a fibrotic tubed pedicle and in case 2 from myositis ossificans. According to several case series, bony ankylosis of the mandible and maxilla has also been found to occur in 30%-43% of cases of NOMA.

show abstract

“…3 Most reported cases were neonates with only five cases being older children and adults. 1,2,8,10,11 However in this case mouth opening was decreasing progressively till age of 14 years.…”

Section: Discussionmentioning

confidence: 62%

Unilateral Acquired Posterior Maxillary Mandibular Syngnathia

Sahu

Roy²,

Kashyap³

2016

BJOHNS

View full text Add to dashboard Cite

show abstract

“…Of the 59 cases, 29 were isolated bony syngnathia with no other congenital anomalies, 4,6,10,13,15 -25 whereas 30 were congenital bony syngnathia with other congenital anomalies. 11,[13][14][15][26][27][28][29] Total 16 cases were demonstrated as bony fusion between the ascending ramus and body of mandible with the zygoma and maxilla. In all 8 cases were isolated bony syngnathia 8,9,11,12,15,24 and 8 cases were complex bony syngnathia.…”

Section: Discussionmentioning

confidence: 99%

Congenital Bilateral Zygomatico-Maxillo-Mandibular Fusion Associated With Gum Fusion

Al-Mahdi

Koppel

Al‐jumaily

et al. 2016

Journal of Craniofacial Surgery

View full text Add to dashboard Cite

A congenial syngnathia is very rare condition. It can be simple mucosal fusion (synechiae), or complete bony fusion (synostosis) between the maxilla or zygoma and the mandible. Fusion of the ascending ramus of mandible to maxilla and zygoma is less common than fusions of the alveolar ridges of the mandible to the maxilla. Bony syngnathia is either isolated or complex in form. There are 59 cases of congenital bony syngnathia reported in the literature: the first report was by Burket in 1936. There are 16 reported cases of zygomatico-maxillo-mandibular fusion. In the reported cases, women expressed the isolated form more commonly whereas men demonstrated a more complex pattern of disease. The authors present another patient of bony syngnathia involving bilateral fusion of the ascending ramus and body of the mandible with the maxillary complex in a young man. Early surgery was performed to release the bony and soft tissue fusion on the eighth day from the baby's birth. A second operation was performed for recurrence when the baby was 2.5 months old. A customized splint, an intense postoperative program of mouth exercises, and close follow-up aims to prevent further refusion.

show abstract