|Year : 2021 | Volume
| Issue : 1 | Page : 1-5
Management of localized juvenile spongiotic gingival hyperplasia: A systematic review
Hani H Mawardi1, Soulafa A Almazrooa1, Heba A Turkstani1, Razan S Balkhair2, Alaa G Almasoudi2, Bushra A Bakhamis2, Lujain Z Azzouz2, Tahani A Alshareef2, Soha E Alsulami2, Sarah A Alsahafi2, Asma A Albarqi2
1 Department of Oral Diagnostic Sciences, King Abdulaziz University, Jeddah, Saudi Arabia
2 Department of Dental Intern, Faculty of Dentistry, King Abdulaziz University, Jeddah, Saudi Arabia
|Date of Submission||03-Jun-2020|
|Date of Acceptance||05-Aug-2020|
|Date of Web Publication||04-May-2021|
Dr. Hani H Mawardi
Department of Oral Diagnostic Sciences, Faculty of Dentistry, King Abdulaziz University, Abdullah Ibnkhashab Street, 21432 Jeddah
Source of Support: None, Conflict of Interest: None
Background: Localized juvenile spongiotic gingival hyperplasia (LJSGH) is an uncommon condition presenting as a well-circumscribed, papillary, and exophytic red soft-tissue lesion commonly on the gingival margin and attached gingiva with distinctive histological features. Up to date, the exact etiology is yet to be determined, while a reactive nature of the disease was suggested. Purpose: The aim of this systematic review is to investigate various LJSGH treatment options for the best outcome. Methods: A search was conducted using PubMed/Medline and Medscape up to April 2019. All English literature on management of LJSGH was included and systematically reviewed for bias and using different levels of elimination by multiple reviewers. The required data from eligible studies were extracted and analyzed. Results: Twelve articles met the inclusion criteria following the Preferred Reporting Items for Systematic Reviews and Meta-analysis statement. In total, 97 cases were treated by surgical excision in which 12 had recurrence within a median follow-up of 29 months. Two cases were treated with cryotherapy, one with photodynamic therapy, and one case with surface cauterization with topical clobetasol all with no reported recurrence. In addition, one case was treated with scaling and chlorhexidine application without significant response. Conclusion: Based on the available evidence, the complete excision of LJSGH using any method may have the most predictable outcome. Further studies are needed to confirm these findings and explore alternative management options.
Keywords: Juvenile, localized, spongiotic gingival hyperplasia, systematic review
|How to cite this article:|
Mawardi HH, Almazrooa SA, Turkstani HA, Balkhair RS, Almasoudi AG, Bakhamis BA, Azzouz LZ, Alshareef TA, Alsulami SE, Alsahafi SA, Albarqi AA. Management of localized juvenile spongiotic gingival hyperplasia: A systematic review. J Dermatol Dermatol Surg 2021;25:1-5
|How to cite this URL:|
Mawardi HH, Almazrooa SA, Turkstani HA, Balkhair RS, Almasoudi AG, Bakhamis BA, Azzouz LZ, Alshareef TA, Alsulami SE, Alsahafi SA, Albarqi AA. Management of localized juvenile spongiotic gingival hyperplasia: A systematic review. J Dermatol Dermatol Surg [serial online] 2021 [cited 2022 May 19];25:1-5. Available from: https://www.jddsjournal.org/text.asp?2021/25/1/1/315345
| Introduction|| |
Localized juvenile spongiotic gingival hyperplasia (LJSGH) is an uncommon condition which was first described in 2007 by Darling et al. It usually presents as a well-circumscribed solitary, papillary, and exophytic red soft-tissue lesion commonly found on the gingival margin and attached gingiva [Figure 1]. Unlike other conditions with similar clinical presentation, LJSGH is not related to dental plaque and does not resolve following routine dental prophylaxis. Furthermore, it tends to be more prevalent in children and young adults with more female predilection. Up to date, the exact etiology of LJSGH is unknown; however, it is believed to be reactive in nature. Other studies have suggested a role for human papillomavirus (HPV) in this condition pathogenesis with limited evidence.
|Figure 1: A clinical example for localized juvenile spongiotic gingival hyperplasia presenting as ulceration and erythema of the gingival margin of the maxillary right lateral incisor|
Click here to view
Considering its clinical features, LJSGH may mimic other gingival conditions such as plaque-induced gingivitis, puberty-related gingivitis, and pyogenic granuloma. Therefore, it is typically diagnosed based on the combination of clinical and histopathological features. Darling et al. described the common microscopic criteria of LJSGH which include epithelial hyperplasia, spongiosis, loss of keratinization, and connective tissue infiltration by lymphocyte, plasma cell, and neutrophils. Several treatment modalities to treat LJSGH have been suggested including surgical excision by scalpel or laser, cryotherapy, mild surface cauterization, followed by topical application of clobetasol, photodynamic therapy (PDT), and scaling and root planning (SC/RP) with chlorhexidine (CHX) mouthwash.,,,, However, a gap in the available literature exists on which LJSGH treatment option to provide the most predictable outcome with least risk for recurrence.
In order to provide the best available evidence, the aim of this study was to systematically review the LJSGH literature on available treatment modalities with the best outcome in relation to recurrence risk.
| Methods|| |
A systematic review of the literature was conducted using two search engines of PubMed/Medline and Medscape database up to April 2019. The electronic search followed predetermined PICO questions to identify eligible articles on treatment modalities for LJSGH and risk for recurrence. The search keywords included: Localized juvenile spongiotic gingivitis, juvenile spongiotic hyperplasia, juvenile spongiotic gingiva, surgical excision, surgical intervention all combined by Boolean search operators “AND” and “OR.”
The systematic review inclusion criteria included English literature, diagnosis of LJSGH based on histopathological examination, human subjects, and all types of study designs (case report, case series, retrospective studies, prospective studies, and randomized clinical trials [RCT]). Exclusion criteria included the following: non-English literature, nonhuman studies, reviews, and observational studies. Eligible studies were reviewed by all co-authors for inclusion in this systematic review using different levels of elimination based on titles, abstracts, followed by article full text. Any disagreement between reviewers was resolved by a group discussion before reaching an agreement.
Using the Preferred Reporting Items for Systematic Reviews and Meta-analysis checklist, assessment for bias for individual and across studies was carried over at the study level. The methodological quality for each study was assessed and pointed out flaws in the design. Data from eligible studies were extracted by co-authors including study characteristics of year, designs, the number of participants enrolled, intervention, and outcome. A summary report was generated, with critical appraisal and literature review.
| Results|| |
The initial literature search yielded 16 articles, in which 12 met the inclusion criteria and were included in this study.,,,,,,,,,,, There were nine case reports, two retrospective studies, and one case series [Figure 2]. In total, there were 51 males and 61 females, mostly Caucasian (50.4%), with the mean age of 12.1 years (range from 5 to 39). The anterior gingiva was the most common site (104/112; 92.86%), followed by the posterior gingiva. Based on the clinical presentation of included cases, the differential diagnosis list included plaque-related gingivitis, pyogenic granuloma, hyperplasia or fibrous hyperplasia, inflammatory lesions including gingivitis and stomatitis, giant cell granuloma, HPV-related lesions (e.g., papilloma, condyloma accuminatum, or verruca vulgaris), and erythroplakia in addition to LJSGH. In order to confirm the diagnosis, tissue samples of all included 112 LJSGH cases were submitted for histopathologic examination, and all showed similar features of an exophytic lesion surfaced by nonkeratinizing, hyperplastic stratiﬁed squamous epithelium with brisk spongiosis, and supporting ﬁbrous connective tissue with increased vascularization and infiltration of inflammatory cells. In addition, there was spongiosis of the spinous layer and neutrophilic exocytosis.
|Figure 2: Flow chart of the literature search process and elimination strategy|
Click here to view
Several treatment options were offered to LJSGH patients as the second line of treatment which included scalpel excision, laser excision, cryotherapy, SC/RP with CHX mouth rinse, mild surface cauterization, followed by topical application of clobetasol ointment and PDT [Table 1]. Scalpel excision was performed in 93.75% (105/112) of all included cases, in which recurrence was reported in 14.29% (15/105) of cases. At the same time, laser excision was offered to 4.46% (5/112) of patients with no reported recurrence up until the median follow-up of 16 months (range 6–24).,,,,,,,,,, In all included cases, excision of LJSGH was conducted once except for five cases where a second revision surgery was needed for lesion recurrence.,,
Cryotherapy was reported in one study, where open-spray technique with liquid nitrogen was delivered to two patients (1.8%). In this technique, topical application of 10% xylocaine spray was applied first, followed by the application of liquid nitrogen using a handheld device of 300-mL capacities for a maximum time of 20–30 s and slow thaw time of 30 s. No recurrence of LJSGH was reported in any of both included cases during the follow-up period (5 and 6 months). SC/RP with CHX mouth rinse was delivered to a single case (0.1%) and was followed up every 3 months. During this period, minimal lesion shrinkage was noted, and gingival contouring and/or laser treatment was advised for the complete resolution which was rejected by the patient.
Laser surface cauterization was used to treat a single LJSGH case (0.1%). The treatment course consisted of two CO2 laser applications (2.5 watts and a pulsed energy beam at 80 Hz) with a paintbrush movement. Following the initial surface cauterization, moderate resolution of the lesion was reported. Therefore, a second cauterization treatment followed using topical application of 0.05% clobetasol propionate ointment. One month follow-up demonstrated the resolution of the lesion with mild “residual inflammation” of the gingival margin which was interpreted as plaque-related but persisted over the following 6 months.
PDT was used for a single LJSGH case (0.1%) and consisted of topical application of 0.1% methylene blue for 5 min over the gingival sulcus and marginal gingiva, followed by low-level laser application using DMC Therapy XT at nine sites within the affected area. The treatment protocol consisted of 12 sessions (3 sessions per week on nonconsecutive days) completed over 4 weeks. At 2-year time point, a slight hyperplasia in the interdental papilla with no other signs of LJSGH was noted. Overall, the median follow-up time for all included cases was 26 weeks. The overall reported recurrence rate after the first intervention was 16.9% (19/112); however, it dropped down to 11.6% (13/112) following the second intervention.
| Discussion|| |
The oral cavity is a host for several benign soft-tissue lesions with various clinical presentations. LJSGH is one, which has been recognized recently and presents as a solitary exophytic soft-tissue lesion commonly noted on the gingival margin and attached gingiva of children and young adults mimicking plaque-related gingivitis in most of the cases. However, LJSGH has been reported in the older population and in more extensive fashion. As such, there has been a suggestion to update the diagnostic term to include spongiotic gingival hyperplasia and eliminate the juvenile component. Due to the information gap in the literature and differences in clinical experience among dental practitioners, LJSGH is most likely underreported representing only 0.069% of 31,469 biopsies collected between 2008 and 2014. In addition, and due to the similarity of clinical feature to other soft-tissue lesions, LJSGH has been misdiagnosed under puberty gingivitis, foreign body gingivitis, and peripheral giant cell granuloma before the histopathological examination.
The exact pathogenesis of LJSGH is unclear and yet to be determined. However, several theories have been suggested including the underlying inflammatory process as a response to specific triggers such as plaque and foreign bodies. In addition, a study by Darling et al. proposed LJSGH to be a developmental disorder and originate from an ectopic junctional/sulcular epithelium in the attached gingiva supported by the presence of similar immunophenotypic markers in LJSGH and junctional epithelium. HPV has also been implicated to play a role in LJSGH pathogenesis. The analysis of LJSGH 21 cases demonstrated overexpression of p16INK4A and intense inflammatory infiltration suggestive for HPV involvement. However, further evidence is needed to confirm these theories.
Considering the frequent presentation of LJSGH on the anterior gingiva in an asymptomatic fashion without response to routine periodontal treatment, esthetics becomes the main patient concern requiring dental intervention even with its benign nature. Several treatment options have been proposed including surgical excision, SC/RP combined with CHX mouth rinse, cryotherapy, CO2 surface cauterization, and PDT. Based on the limited current data, surgical excision demonstrated a high success rate with the least recurrence risk. The application of CO2 surface cauterization, followed by topical application of 0.05% clobetasol propionate ointment demonstrated the complete resolution of the lesion in a single case similar to cryotherapy. However, due to low number of reported cases with short follow-up, it will be hard to generalize the treatment outcome for both CO2 and cryotherapy. The application of PDT has demonstrated promising and conservative approach with no reported recurrence up to 2 years of follow-up. Similar to CO2 and cryotherapy, there has been only a single reported case which requires further investigation to confirm this finding.
This systematic review has several limitations. First, only 12 articles met the inclusion criteria and were mostly case series and case reports which account for a low level of scientific evidence. Second, the reported follow-up period for several studies was more on the short side. Third, due to lack of reported details, it is challenging to extrapolate if all surgical excision procedures were performed correctly to excise the LJSGH lesion without remnants which may contribute to the recurrence rate. Therefore, a lack of RCTs with longer follow-up may not allow for generalization of the study outcomes.
| Conclusion|| |
LJSGH is a fairly new disease, in which most of the clinicians in the dental field are not aware of compromising the dental care delivered to patients. Based on the included studies, the excision of LJSGH whether by scalpel or laser may demonstrate a predictable treatment outcome compared to other treatment options. Future RCTs are needed to establish an evidence-based management approach for LJSGH.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Darling MR, Daley TD, Wilson A, Wysocki GP. Juvenile spongiotic gingivitis. J Periodontol 2007;78:1235-40.
Argyris PP, Nelson AC, Papanakou S, Merkourea S, Tosios KI, Koutlas IG. Localized juvenile spongiotic gingival hyperplasia featuring unusual p16INK4A labeling and negative human papillomavirus status by polymerase chain reaction. J Oral Pathol Med 2015;44:37-44.
Chang JY, Kessler HP, Wright JM. Localized juvenile spongiotic gingival hyperplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;106:411-8.
Nogueira VK, Fernandes D, Navarro CM, Giro EM, de Almeida LY, León JE, et al
. Cryotherapy for localized juvenile spongiotic gingival hyperplasia: Preliminary findings on two cases. Int J Paediatr Dent 2017;27:231-5.
Flaitz CM, Longoria JM. Oral and maxillofacial pathology case of the month. Localized juvenile spongiotic gingival hyperplasia. Tex Dent J 2010;127:1312-3, 1315-7.
Vieira DL, Leite AF, de Souza Figueiredo PT, Vianna LM, Moreira‐Mesquita CR, de Melo NS, et al. A conservative approach for localized spongiotic gingivitis hyperplasia using photodynamic therapy: A case report and review of the literature. Photobiomodul Photomed Laser Surg 2019;37:57-61.
MacNeill SR, Rokos JW, Umaki MR, Satheesh KM, Cobb CM. Conservative treatment of localized juvenile spongiotic gingival hyperplasia. Clin Adv Periodontics 2011;1:199-204.
Petruţiu ŞA, Roman A, Soancă A, Sârbu C, Stratul ŞI. Localized juvenile spongiotic gingival inflammation: A report on 3 cases. Clujul Med 2014;87:198-202.
Kalogirou EM, Chatzidimitriou K, Tosios KI, Piperi EP, Sklavounou A. Localized juvenile spongiotic gingival hyperplasia: Report of two cases. J Clin Pediatr Dent 2017;41:228-31.
Solomon LW, Trahan WR, Snow JE. Localized juvenile spongiotic gingival hyperplasia: A report of 3 cases. Pediatr Dent 2013;35:360-3.
Siamantas I, Kalogirou EM, Tosios KI, Fourmousis I, Sklavounou A. Spongiotic gingival hyperplasia synchronously involving multiple sites: Case report and review of the literature. Head Neck Pathol 2018;12:517-21.
Oupadissakoon C, Singhatanadgit W, Chaiprakit N. Localized juvenile spongiotic gingival hyperplasia: A case report. CU Dent J 2016;39:95-100.
[Figure 1], [Figure 2]