From Volume 47, Issue 2, February 2020 | Pages 162-164
Localized juvenile spongiotic gingival hyperplasia is a relatively new histopathological diagnosis, presenting as a localized erythematous lesion of the anterior attached gingivae. Presenting in a peri-pubertal age, this condition is often misdiagnosed as ‘puberty gingivitis’. Here, two cases presenting to the Joint Paediatric-Oral Medicine clinic at the Eastman Dental Hospital are discussed.
Localized Juvenile Spongiotic Gingival Hyperplasia (LJSGH) is a relatively new histopathological diagnosis, being first described in 2007.1 Clinically, LJSGH distinctively presents as erythematous, raised papillary lesions affecting a discrete area of attached gingiva which, importantly for diagnostic purposes, is often independent of the unaffected adjacent marginal gingiva. These lesions are most commonly noted on the labial aspect of the anterior maxilla, as seen in approximately 81% of cases, however, anterior mandibular involvement has also been reported.2
Macroscopically, these exophytic lesions have a granular or ‘pebble-like’ appearance, and are often painless. Bleeding on brushing is the only reported symptom in approximately 20% of cases. Consequently, delayed clinical presentation is often a feature of this condition. LJSGH presents in a peri-pubescent age group, with a female predominance reported.3 Given this presentation, LJSGH is often erroneously diagnosed as ‘puberty gingivitis’. It is refractory to mechanical periodontal treatment, which differentiates it from other gingival conditions.4 Microscopically, the presence of acanthotic stratified squamous epithelium, with intercellular oedema and epithelial infiltration of inflammatory cells, namely neutrophils, is diagnostic. There is also marked differentiation from peri-lesional normal mucosa.5
A 10-year-old boy was referred to the Joint Paediatric-Oral Medicine clinic with a five-month history of an erythematous ‘spongy’ lesion involving the gingiva of the upper left central incisor. The patient reported no discomfort but occasional bleeding on brushing was noted. There was no improvement following improved oral hygiene. The medical history was unremarkable.
On examination, a 6 mm diameter pedunculated, granular lesion extending from the attached gingiva of the upper left central incisor to the muco-gingival junction was noted (Figure 1). The marginal gingiva was uninvolved. The lesion did not blanch on touch. There was no bleeding on probing or periodontal pocketing. Hypomature Amelogenesis Imperfecta (AI) was noted.
An excisional biopsy was performed, with the area left to heal by secondary intention. Histopathology demonstrated mild intercellular oedema and infiltration by neutrophils consistent with the diagnosis of LJSGH. Review at three months showed no evidence of recurrence, and intact gingival architecture.
A 10-year-old girl was referred with a one-year history of a gingival swelling involving the upper left lateral incisor. There was a history of bleeding on brushing but the patient was otherwise asymptomatic. The medical history was unremarkable.
On examination, a 4 mm pedunculated, velvety, erythematous lesion was noted on the attached gingiva of the upper left lateral incisor (Figure 2). The lesion was non-blanching. There was no bleeding on probing or periodontal pocketing. There was no interval improvement following subgingival debridement. An excisional biopsy was performed with chemical cauterization to achieve haemostasis at the gingival margin (Figure 3).
Histological findings were consistent with a diagnosis of LJSGH, confirming intercellular oedema and neutrophilic infiltration. At three month review, there was mild erythema confined to the gingival margin only with no evidence of recession (Figure 4).
Localized juvenile spongiotic gingival hyperplasia presents as erythematous, raised papillary lesions affecting a discrete area of attached gingiva. The differential diagnosis for isolated exophytic gingival lesions includes pyogenic granuloma, peripheral ossifying fibroma, benign vascular lesions or a peripheral giant cell granuloma.6,7 The diagnosis should be suspected in a peri-pubertal patient presenting with the characteristic clinical features.
The aetiology of LJSGH remains unknown, however, multiple hypotheses have been suggested, including trauma, lip incompetence and placement of orthodontic appliances. These are, however, likely to be confounding factors given the typical age of presentation.8 The peri-pubertal presentation of LJSGH has also given rise to the suggestion of a hormonal aetiology. Immunohistochemical studies for the presence of oestrogen and progesterone receptors have been negative.1 Despite a papillary phenotype, LJSGH does not present with the characteristic histopatholologic features of HPV.8 There is now growing evidence to suggest that LJSGH represents ‘exteriorization’ of the junctional epithelium, which has been demonstrated by similarity in the immunohistochemical staining patterns of expressed cytokeratins between these two tissues.1,5 It is hypothesized that the ectopic junctional epithelium becomes inflamed and hyperplastic in response to irritation and mechanical trauma, which results in the distinctive clinical features of LJSGH. Importantly, LJSGH is not a plaque-related process, and is clinically distinct from puberty gingivitis, proving refractory to oral hygiene measures, with no hormonal influence and no marginal gingival involvement.9
Whilst spontaneous resolution has been suggested, there have been no prospective studies to support this as a conservative management option currently.4 While literature on management is sparse, this condition is unresponsive to topical corticosteroids,10 with surgical excision currently the favoured treatment modality. This may be associated with iatrogenic risk and disruption of local gingival architecture in a highly aesthetic zone. A recent study has shown positive treatment outcomes in two cases using cryotherapy.11 Recurrence rates between 6% and 16% have been documented following surgical excision, with follow-up varying from one month to five years.3
Localized juvenile spongiotic gingival hyperplasia is an under-reported condition, often being erroneously misdiagnosed as pubertal gingivitis, largely attributed to its presentation in a peri-pubertal age group. Further research is required to confirm the aetiology and pathogenesis of this condition, and long-term follow-up studies to establish the incidence of recurrence post-treatment.
