Unusual presentation of a tooth in a 9-year-old child with cleft lip and palate: a case report

Cleft lip and palate is the most common craniofacial malformation with a reported worldwide incidence of 1.7 per 1000 live births.² While the severity of each individual cleft can vary, the principles behind the treatment are the same. In the United Kingdom, primary lip repair is undertaken at around 3 months of age, with affected palates undergoing primary repair at approximately 9 months.

Ectopic teeth are not uncommon in patients who have cleft lip and palate. In the general population, the ectopic development of teeth has been reported in a number of locations around the oral cavity including the palate,³ maxillary sinus,⁴ condyle,⁵ coronoid process,⁶ orbital floor⁷ and nasal cavity.⁸

Nasally erupting teeth can be due to trauma,⁹ cleft lip and palate,¹⁰ or idiopathic causes.¹¹ They are an uncommon complication of cleft lip and palate and, subsequently, there are only a few reported cases in recent literature.¹⁰

The aetiology behind cleft lip and palate associated intranasal teeth is not fully understood owing to the limited number of well-documented cases. It has been hypothesized that, during the incomplete formation of embryonic processes, a tooth germ can be displaced from its ideal location. This can subsequently be further malpositioned by extensive surgical procedures, and result in ectopic eruption into the nasal cavity.¹²

Intranasal teeth are occasionally asymptomatic¹² and discovered as an incidental finding following routine clinical and radiological investigations. More frequently, they present with a diverse range of clinical symptoms, including nasal obstruction,¹³ congestion¹⁴ and discharge,¹⁵ recurrent epistaxis,¹⁶ and pain.¹⁷ The teeth themselves can be deciduous, permanent or supernumerary.^8,12,14

The treatment of intranasal teeth involves their removal to alleviate symptoms and reduce the potential for further complications. This is followed by cosmetic surgery, if required.¹⁷ In one reported case, the intranasal tooth was left in situ. The literature suggests that, in these instances, routine radiological monitoring should be undertaken.^11,14

Case report

The subject of this case report was a 9-year-old female with a right-sided unilateral cleft lip and palate, under the care of the orthodontic department at St Luke's Hospital, Bradford. Her previous treatment had involved surgical repair of the right-sided unilateral cleft lip at 4 months of age and then the cleft palate at 10 months. She had been attending the orthodontic department over the previous 15 months, for both arch expansion and the use of a sectional fixed appliance in the upper arch to reposition a malpositioned and distally inclined UR1, which was causing labial ulceration, and to create space in preparation for an alveolar bone graft. The patient reported a new complaint of a tooth growing into her nasal cavity. The surrounding nasal area was tender to touch and nose wiping was reportedly sore.

Clinically, an object which appeared to be the crown of a tooth could be seen up the patient's right nostril (Figures 1 and 2). While an OPG radiograph (Figure 3) taken was diagnostically inconclusive of an intranasal tooth, a lateral cephalogram (Figure 4) clearly showed the presence of two odontome-like structures positioned in the nasal cavity.

The patient was reviewed at the subsequent cleft lip and palate clinic with the cleft surgeon, where it was decided to remove the supernumerary teeth under general anaesthetic via a nasal approach.

This case illustrates that, particularly in cleft lip and palate patients, teeth may present in unexpected positions. It also highlights the value of taking two radiographs, at a 90° angle to each other, to aid the localization of ectopic teeth; as well as demonstrating that panoramic radiographs do not always clearly show the presence of ectopic teeth that lie outside the focal trough.