Iatrou I, Theologie-Lygidakis N, Leventis M. Intraosseous cystic lesions of the jaws in children: a retrospective analysis of 47 consecutive cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009; 107:485-492 https://doi.org/10.1016/j.tripleo.2008.10.004
Tkaczuk AT, Bhatti M, Caccamese JF Cystic lesions of the jaw in children: a 15-year experience. JAMA Otolaryngol Head Neck Surg. 2015; 141:834-839 https://doi.org/10.1001/jamaoto.2015.1423
Patel M, Schultz K, Rosenfeld E. The earliest known reported occurrence of dentigerous cyst in a six-month-old child. Int J Oral Maxillofac Surg. 2022; https://doi.org/10.1016/j.ijom.2022.06.014
Robinson RA. Diagnosing the most common odontogenic cystic and osseous lesions of the jaws for the practicing pathologist. Mod Pathol. 2017; 30:(s1)S96-S103 https://doi.org/10.1038/modpathol.2016.191
Gallichan N, Albadri S, Dixon C, Jorgenson K. Trends in CBCT current practice within three UK paediatric dental departments. Eur Arch Paediatr Dent. 2020; 21:537-542 https://doi.org/10.1007/s40368-020-00526-w
Sahu B, Anand R, Kumar S A pattern-based imaging approach to pediatric jaw lesions. Indian J Radiol Imaging. 2021; 31:210-223 https://doi.org/10.1055/s-0041-1729767
Cystic lesions of the jaw in the paediatric population have been infrequently documented in the literature. The majority of these lesions are considered to be developmental in nature, as opposed to the commonly noted inflammatory lesions in the adults. Although asymptomatic, these lesions are associated with a high level of anxiety for parents. Early diagnosis and management help to allay anxiety. We present a case of 6-year-old boy who had a rapidly growing lucent lesion of the mandible that needed urgent intervention. The lesion was excised, analysed histopathologically, and confirmed as a dentigerous cyst. The management of paediatric cystic lesions can be difficult and vary significantly from those of the adult population. This can be amplified because cross-sectional imaging is seldom used in children owing to the risk associated with radiation. We present our experience of dealing with rapidly progressing lesion and the lessons learned in the process.
CPD/Clinical Relevance: The management of paediatric cystic lesions can be difficult and vary significantly from those of the adult population.
Article
A 6-year-old boy presented to the department of oral and maxillofacial surgery on an urgent pathway by his GP. There was a history of asymptomatic, but progressive swelling of the jaw for the previous 3 months. His past medical history was insignificant, and specifically there had been no weight loss, night sweats or pyrexia.
On examination, there was no obvious cervical lymphadenopathy. A visible and hard right-sided facial swelling was noted, with no overlying skin changes. Intra-oral examination revealed expansion of mandible around the LRC–LR6 region, with LR DE missing. There was no associated tenderness, discharge, ulceration or lesion on the mucosa. An orthopantogram (OPG) was taken at this visit, which showed a 36 x 25 mm, unilocular, well-defined radiolucency of the right mandible, extending from the apex of the deciduous canine to the first molar region. Additionally, the border of mandible adjacent to the lesion was very thin and susceptible to pathological fracture (Figure 1). In view of the above, an urgent surgery was planned to either marsupialize or enucleate the lesion under general anaesthesia.
A standard mucoperiosteal flap was raised around the lesion to visualize the mandibular expansion. The cyst was completely enucleated, with removal of the partially developed hypoplastic LR5, which was anchored to the cyst (Figure 2). All adjacent structures, including the mental nerve and the thin mandibular border were protected throughout the procedure. The patient was prescribed a soft diet for 6 weeks after the operation. Histological analysis confirmed that the lesion was a dentigerous cyst. The patient was followed up 6 weeks post-operatively, and he showed satisfactory healing, with no pain or paraesthesia in the surrounding tissue.
Discussion
Cystic lesions are uncommon and generally indolent in children. They are usually detected on routine radiological surveillance by dental practitioners. In children, developmental cysts, such as dentigerous and odontogenic keratocysts are more common than the inflammatory cysts noted in adults.1 Tcakzuk et al, in a 15-year study of cystic lesions of the jaw in a large university hospital found only 57 paediatric patients with true cystic lesions.2 Some unerupted teeth are associated with dome-shaped fluid-filled lesions that regress after eruption of the associated tooth. These are called eruption cysts and are most commonly noted in the maxillary region in the central incisor and molar regions.7 Dentigerous cysts, on the other hand, are formed by fluid accumulation between the reduced enamel epithelium and the crown during development, and are usually noted between the second and third decades of life, with the earliest presentation in literature seen in a 6-month-old infant.3 On the panoramic radiograph, they typically present as a unilocular, radiolucent lesion that is attached to the cemento-enamel junction (CEJ) of an unerupted or impacted tooth.4
On review of imaging, pathological lesions of the jaw are commonly divided into radiolucent, radiopaque and mixed-density lesions. Radiolucent lesions of the mandible in the paediatric population are generally benign and non-odontogenic in nature, the most common being solitary bone cysts, aneurysmal bone cysts, central giant cell granuloma and Langerhans cell histiocytosis. Among the benign, odontogenic lucent lesions, radicular cysts, dentigerous cysts, keratocystic odontogenic tumours, ameloblastoma and odontogenic myxoma are encountered in children. Malignant entities, such as Ewing's sarcoma and lymphoma, should always be considered in the surgical sieve when diagnosing radiolucent lesions of the jaw.6
Although, cross-sectional imaging was not requested in this case, there is a distinct role for special imaging owing to its beneficial role in risk-assessment and surgical planning for management of lucent lesions in general. Cone beam computed Tomography (CBCT) has proven to be an important tool in the diagnosis of various conditions of the maxillofacial region. However, its use is limited in paediatric population owing to worries regarding increased radiosensitivity of tissues in children and its potential carcinogenic effects. A three dental hospital study over a 4-year period in UK revealed a mean age of CBCT exposure to be 11 years.5 The use of CBCT has increased in children, especially when a lower field of vision (FOV) is examined, thus reducing effective radiation. Nevertheless, a careful risk versus benefit assessment should be carried prior to prescribing cross-sectional imaging in children.
Conclusion
It is both unusual and worrying for parents to see their children showing expansile lesions of the jaw. Early diagnosis and intervention are paramount in these cases. We present a case of a mandibular dentigerous cyst in a 6-year-old patient, which is very seldom seen at this age, and its management for the benefit of general dentists and oral surgeons in the community.