Abstract
Introduction
Congenital maxillomandibular syngnathia, or fusion of the jaws, is a rare condition that has a broad spectrum of presentations. The restricted mouth opening can lead to issues with feeding, swallowing, and respiration resulting in failure to thrive and temporomandibular joint ankylosis. Early recognition and treatment is necessary for proper growth and development.
Case report
We report a 1-day-old male with isolated bilateral soft tissue alveolar fibrous bands. He presented with difficulty feeding secondary to trismus. No bony or muscular involvement in the synechiae was noted and the remainder of the physical exam was unremarkable. The bilateral alveolar synechiae were divided under local anesthesia using surgical scissors. The patient immediately showed improvement in mouth opening and had resolution of his feeding problems. He is now gaining weight and developing appropriately.
Discussion
The accompanying review of the literature demonstrates only 11 cases worldwide of isolated maxillomandibular fusion. Depending upon the composition of the synechiae, simple surgical division under local anesthesia can be curative.
1
Introduction
Congenital maxillomandibular syngnathia, first reported in 1936, is a rare deformity leading to difficulties in feeding and ability to thrive. The presentation may be unilateral, bilateral, or complete, but is most often syndromic in etiology . Isolated cases of maxillomandibular fusion are extremely rare. The composition of the fusion can also vary from mucosa only to muscle and/or bony involvement. If the treatment is delayed, ankylosis of the temporomandibular joint may result . A case report of a baby boy with isolated bilateral maxillomandibular fibrous bands is presented. The included review of the literature demonstrates only 11 cases worldwide of isolated maxillomandibular synechiae.
2
Case report
A baby boy, born via an uncomplicated vaginal delivery, was noted to have difficulty feeding due to trismus. The baby’s mouth opening was approximately 6 mm. Upon examination, it was established that the trismus was a result of bilateral posterior maxillomandibular fibrous bands at the approximate location of the primary molars ( Fig. 1 ). The bands were estimated to be 1 cm long and 2 mm wide. It was clear that the bands did not contain any bony component due to the easily compressible presentation ( Fig. 2 ). No other craniofacial abnormalities were noted on exam; however, a complete palate exam was limited by the trismus. The mandible was palpated along with the temporomandibular joint and no obvious abnormalities were noted. For the first day of life, the baby was fed using a syringe and did not display any other difficulties.
The mother had an uncomplicated pregnancy and received proper prenatal care. The family history was also unremarkable. This baby boy was the family’s first child.
Following injection of local anesthetic (1% lidocaine with 1:100,000 epinephrine) into the fibrous bands, the adhesions were divided using surgical scissors. The procedure was tolerated well by the patient and minimal blood loss was noted. The mouth opening was immediately improved to approximately 1.5 cm; however, a minimal amount of restriction was still present. The palate was noted to be intact and the baby had a good suck reflex. The baby was discharged later in the day after demonstrating the ability to breastfeed without difficulty.
At a 1-week follow-up appointment, the baby was noted to be doing well. No reports of difficulty feeding were expressed and he was starting to gain weight appropriately. The mouth opening was still believed to have minimal restriction; however, given the baby’s improvement, further evaluation was delayed ( Fig. 3 ).
