Abstract
An extensive sublingual ulceration in a two year old girl with delayed development was reported. The severe lesion resulted from repetitive trauma caused by the lower central incisors in combination with uncontrolled movement of the orofacial muscles. Treatment successfully healed the lesion, however an deformity of the tongue remained afterwards. Given this risk, health care providers should be aware of the danger posed by sublingual ulcerations in children with neurologic conditions when the child’s lower primary incisors erupt. Early diagnosis and referral to a pediatric dentist can prevent progression of the lesion as well as deformity of the tongue.
Sublingual ulceration is a rare type of oral lesion usually found in infants up to two years old. This lesion is characterized by a round red border with a yellowish slough in the middle. The size of the lesion varies from small to very large (2–30 mm. in diameter), depending on the lesion’s cause and age . Most sublingual ulcerations are caused by abrasion from natal or neonatal teeth, and when the condition occurs in the first year of life, it is also referred to as Riga-Fede disease . When these ulcerative lesions are found in patients older than two years old, they are called oral traumatic granulomas or simply sublingual ulcerations .
The complaint most commonly bringing the affected infant to a dentist is pain or feeding difficulty. Conservative treatments include grinding the incisal edges of neonatal teeth as well as placement of composite. In more invasive treatment, tooth extraction usually resolves the issue and relieves the pain, when conservative treatment is ineffective.
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Case report
A 2-year, 1-month old girl was referred from University Hospital to Dental Hospital because of a large oral ulceration on the tongue’s ventral surface. Her mother explained that the lesion had appeared a few months ago and since then grown steadily larger. The mother was worried about active bleeding that occurred at night and during daytime naps. Although the girl was yet unable to speak, she showed clear signs of discomfort, including poor mood, irritability, and crying.
The child was born full term with normal weight but with birth asphyxia and hypoxic ischemic encephalopathy. Her medical history included recurrent pneumonia and many long hospitalizations. Development of both gross and fine motor skills was also delayed, including inability to sit up or stand up on her own or walk. Upper and lower limbs were hypotonic. The girl had moderate hearing loss and bilateral otitis media effusion. She had a myringotomy with papurella tube insertion at age 1-year, 10-months. Eye fundi were normal, but visual assessment was impossible. The child received daily oral anti-epileptic medication (Vigabatrin and Phenobarbital) and anti -GERD medication. The patient was fed via percutaneous endoscopic gastrostomy (PEG) and could also suck milk from a bottle without aspiration. She was training with an occupational therapist regularly.
Extraoral examination confirmed that the girl was underweight; unable to sit, stand, or walk; unable to communicate and that her development was delayed. She was totally dependent on her caretaker. The child’s arms and legs were spastic, particularly when awake, and her voluntary movements were often jerky. Her weight was only 8 kg. She also looked pale and weak.
Intraoral examination showed an ulcerative lesion of 5 × 15 mm on the tongue’s ventral surface and extending to the tip. The lesion was an actively bleeding ulcerated area with a yellowish white slough in the middle and a raised border ( Fig. 1 ). The tip of the tongue was physically split by the ulceration. The opposing two primary lower central incisors had normal mobility with slightly sharp incisal edges. The mother reported a history of repetitive trauma, with the mandibular teeth hitting the tongue during the child’s spastic or jerky movement. The lesion caused the girl pain when using the bottle. Sometimes pain from the repetitive trauma woke her up at night, and she cried. This ulcerative lesion concerned the Pediatric Unit, and thus the child was referred to the Dental Hospital. After the dental examination, the pediatric dentist spoke with the mother about possible options for treatment as well as for behavior management. It was decided to first try conservative treatment. The child was held in place by the mother on a dental chair. A mouth gag was placed to keep the child’s mouth open. The dentist used a low speed white stone bur to reduce sharpness of the incisal edges of the primary lower central incisors. The operation took only few minutes. To reduce discomfort from the lesion, triamcinolone acetonide gel (0.1%) was prescribed to apply to the ulcerative lesion twice daily: once in the morning after eating, and again before bedtime. After the operation, the child was scheduled for a follow up appointment one week later. At that follow up, the sublingual lesion looked much better. No active bleeding was found. The child also looked happier than before. The mother reported that the child was able to use her bottle normally again without crying. The girl was also able to sleep well all night. The primary lower central incisors had normal color and mobility. At a second follow up appointment two weeks after the operation, the sublingual ulceration had clearly healed further and was continuing to do so. No sign or symptom of infection or inflammation was found. One month after the operation, the lesion was completely healed. Unfortunately structural deformity remained at the tip of the tongue in the form of bifid tongue ( Fig. 2 a). The patient’s mother reported that the child could feed normally. At the 1 year follow up visit (no 6 month follow-up was possible), there was still no sign of recurrent inflammation, although the tongue deformity persisted. In this case of bifid tongue, there was a clearly visible curving gap at the tongue’s tip, where tissue had been lost ( Fig. 2 b,c). Following up with the patient 1.5 and 2 years after treatment, there was still no recurrent ulceration, even after the upper and primary lower lateral incisors had erupted ( Fig. 2 d,e).
