Publications
Atraumatic intraoral buccal fat pad herniation in a neonate
Clyde Richard Menezes, Rajkiran Raju , Deepa Susan John,1 Shubha Mahadevaiah SUMMARY An approximately 2-day-old neonate was brought with a fleshy intraoral mass and an inability to suckle adequately, without a preceding history of trauma. Contrast-enhanced CT revealed a fat density mass-like lesion occupying and partially filling the oral cavity. Following detection of a defect in the buccinator muscle with associated herniation of the buccal pad of fat into the mouth, a surgical approach was adopted to tackle the condition. Intraoperatively, the defects in the buccinator muscle and buccal mucosa were repaired after excision of the prolapsed fat pad. Age-appropriate feeding practices were initiated following surgery without residual anatomical or functional limitation. Early radiological diagnosis directing surgical intervention was helpful in management of a benign but functionally impeding lesion. BACKGROUND Intraoral masses in neonates are rare, yet have diverse aetiologies.1 2 While herniation of buccal fat through the buccinator muscle has been reported, many of these are secondary to trauma and seen more often in older children.3–6 The exact aetiology for atraumatic herniation, however, still remains elusive.7 When occurring in neonates, mass lesions within the oral cavity can impair proper feeding, compromise the airway, predispose to infection and result in further morbidity.3 5 6 Accurate diagnosis using non-invasive imaging studies is, therefore, CASE PRESENTATION A male baby, born via a normal vaginal birth, was brought to hospital at 2days of life with a fleshy mass within the mouth. The mass, as noticed by the parents, was initially the size of a peanut but increased rapidly within the span of a day to occupy the entire right half of the mouth, hindering feeding. There was no history of facial injury in the intrapartum or postpartum period, with no history of respiratory distress. On examination, the baby was active with stable vitals, no facial asymmetry or dysmorphism. A paleyellow fleshy mass measuring about 3×2cm was seen above the surface of the tongue arising from the right upper jaw. A nasogastric tube was inserted as the lesion prevented proper suckling. Due to the fleshy nature of the mass, a presumptive diagnosis of a mandibular epulis was made. INVESTIGATIONS Contrast-enhanced CT imaging was deemed the most appropriate imaging modality due to its rapidity, ease of performance in a neonate and non-requirement of sedation. Imaging of the face revealed a fat density lesion extending from the region of the right buccal fat pad, protruding through a defect in the ipsilateral buccinator muscle, to lie above the tongue (figures 1 and 2). With a presumptive diagnosis of buccal fat herniation, parental consent was obtained and surgery performed. DIFFERENTIAL DIAGNOSIS In keeping with the fleshy nature of the lesion on examination, a clinical diagnosis of epulis was considered. DISCUSSION Intraoral lesions in neonates are a cause of anxiety to parents; a diagnostic challenge to the treating team considering varied pathologies with similar presentation and may be of concern, especially when associated with respiratory insufficiency or feeding difficulty.8 The exact aetiopathogenesis, however, remains unclear. The first description of an intraoral lipoma was by Roux in 1848, which he referred to as ‘yellow epulis’ denoting mature adipose tissue.9 Furlong believed these lipomas to be part of a spectrum of oral peripheral mesenchymal tumours and proposed a classification10 which was later simplified into diffuse, encapsulated and superficial variants.9 Though buccal mucosa and the buccal alveolar sulcus are the most common sites of involvement, the lips, tongue, soft palate, alveolar ridge and mental regions may also be rarely involved.11 Atypical variants of these intraoral lipomas in older children and adolescents include angiolipomas and pleomorphic subtypes. These are usually larger, more vascular and lobulated as compared with lipomas and have a propensity for infiltration into deeper structures including muscle and bone.12 Classical buccal fat-pad herniation has been reported rarely in infants and preschool toddlers. Typical presentation is with an intraoral mass occasionally accompanied by signs of secondary infection or inflammation.7 The preponderance in this age group may be explained by prominent buccal pads of fat and the predominant tendency of placing objects in mouth resulting in unwitnessed injuries. Bilateral involvement, though exceedingly rare, has still been reported.13 Alternate hypotheses postulated include congenital weakness in the parotid-masseteric fascia described by Matarasso14; traumatic perforation of the buccinator muscle and fascia; neuronal hypoplasia with facial asymmetry, as in geriatric cases; or inherent weakness in the immature musculoskeletal system.15 In the report presented here, we believe that a defect in the buccoalveolar ridge may have resulted from congenital dehiscence in attachment of bucco-pharyngeal fascia resulting in herniation of the buccal fat on vigorous suckling. The common differential diagnoses of intraoral pedunculated swellings of the cheek, in neonates, include lipoma, pyogenic granuloma, neurofibroma, traumatic fibroma, haemangioma, granular cell tumour, cavernous angioma and sialolithiasis of the parotid gland.2 16 Appropriate imaging augments the diagnostic capability of a thorough clinical examination. While MRI is ideal for delineating soft-tissue masses and their locoregional extension, CT imaging boasts greater diagnostic yield in children stemming from the ease of performance, rapidity of image acquisition, minimisation of movement artefacts and better demonstration of calcification and vascularity within masses. Treatment entails surgical excision of the prolapsed buccal fat with closure of the defect in the musculature. Short-term and long-term outcomes are excellent with no late recurrences
