UNIVERSIDADE FEDERAL DE SANTA CATARINA
CENTRO DE CIÊNCIAS DA SAÚDE
DEPARTAMENTO DE ODONTOLOGIA
CURSO DE GRADUAÇÃO EM ODONTOLOGIA
Letícia Machado Berretta
Efetividade das técnicas de marsupialização e descompressão na redução de lesões císticas dos maxilares: uma revisão sistemática
Florianópolis
Letícia Machado Berretta
Efetividade das técnicas de marsupialização e descompressão na redução de lesões císticas dos maxilares: uma revisão sistemática
Trabalho Conclusão do Curso de Graduação em Odontologia do Centro de Ciências da Saúde da Universidade Federal de Santa Catarina como requisito para a obtenção do título de Bacharel em Odontologia.
Orientadora: Profa. Drª. Elena Riet Correa Rivero.
Coorientador: Msc. Gilberto de Souza Melo.
Florianópolis
Letícia Machado Berretta
Efetividade das técnicas de marsupialização e descompressão na redução de lesões císticas dos maxilares: uma revisão sistemática
Este Trabalho Conclusão de Curso foi julgado adequado para obtenção do Título de Cirurgiã-Dentista e aprovado em sua forma final pelo Curso de Graduação em Odontologia da
Universidade Federal de Santa Catarina.
Florianópolis, 22 de outubro de 2019.
________________________ Profa. Dra. Glaucia Santos Zimmermann
Coordenadora do Curso
Banca Examinadora:
________________________ Profa. Dra. Elena Riet Correa Rivero
Orientadora
Universidade Federal De Santa Catarina
________________________ Profa. Dra. Carolina Amália Barcellos Silva
Avaliadora
Universidade Federal De Santa Catarina
________________________ Prof. Dr. Luiz Fernando Gil
Avaliador
Dedico este trabalho à minha família, por todo amor e incentivo que tive para a realização dos meus sonhos. Sem vocês, eu nada seria.
AGRADECIMENTOS
Aos meus pais, Ewandro e Marilisa, por todo amor, zelo, confiança e carinho que me deram ao longo da vida. Agradeço por terem proporcionado meu crescimento pessoal e profissional ao permanecer longe de casa e por me incentivaram a não desistir frente aos obstáculos desta jornada. Muito obrigada pela compreensão dos medos e inseguranças que apresentei e, por tantas vezes, terem acreditado mais em meu potencial do que eu mesma. Vocês são meus maiores exemplos. Obrigada por tudo.
À toda minha família, em especial aos meus irmãos, Ewandro Filho e Bianca, e à minha avó, Ester, por todos os conselhos, pelo companheirismo e por terem sempre me apoiado ao longo destes últimos anos.
Ao meu namorado, Eduardo, por ter sido meu alicerce durante estes cinco anos de graduação. Obrigada pelo apoio imensurável, pelo incentivo ao meu crescimento e por tornar minha vida muito mais leve.
À minha dupla de faculdade, Helena, por ter me ensinado a ser mais confiante e por ter permanecido ao meu lado ao longo das adversidades que enfrentamos. Agradeço por termos crescido juntas, desde o primeiro dia de aula.
À minha orientadora, Elena Riet Correa Rivero, pela oportunidade e confiança para a realização deste trabalho. Muito obrigada por todos os ensinamentos que me foram passados. Sinto-me honrada em ser sua orientada.
Ao meu coorientador e amigo, Gilberto de Souza Melo, por todo auxílio e incentivo na execução deste trabalho e ao longo da minha graduação. Muito obrigada pela sua paciência ao ensinar e pela confiança em meu potencial.
A todos os professores excepcionais que tive a honra de conviver durante a minha graduação, àqueles que são encantados pela odontologia e motivados pela docência. Agradeço pela inspiração pessoal e profissional.
À Universidade Federal de Santa Catarina, por permitir minha formação muito além da profissional. Sou muito grata por tudo que vivenciei nesta instituição e por ter me tornado um ser humano melhor para o mundo. Tenho orgulho de carregar comigo o nome UFSC.
APRESENTAÇÃO
Esta revisão sistemática foi originalmente escrita em formato de artigo na língua inglesa, a fim de ser submetida ao periódico Clinical Oral Investigations, sendo realizada em parceria com os pesquisadores Profa. Dra. Elena Riet Correa Rivero, Msc. Gilberto de Souza Melo, C.D. Fernanda Weber Mello da Universidade Federal de Santa Catarina e o pesquisador Prof. Dr. Giuseppe Lizio da Universidade de Bolonha/Itália.
RESUMO
O objetivo desta revisão sistemática foi avaliar a efetividade das técnicas cirúrgicas de marsupialização e descompressão na redução de lesões císticas dos maxilares. Foram elaboradas estratégias de busca para seis bases de dados eletrônicas e três bases da literatura cinzenta. Foram considerados elegíveis os estudos que avaliaram a efetividade dessas terapias em relação à redução das lesões císticas, após a execução dos tratamentos. Foram identificadas 1298 referências até maio de 2019, das quais 27 estudos quasi-experimentais foram incluídos. Nove estudos avaliaram a técnica de marsupialização e dezoito de descompressão. Uma alta heterogeneidade foi observada entre os estudos em relação às medidas de desfecho, como porcentagem de redução, classificação da Nakamura, tempo de meia-vida, velocidade de redução e aumento na densidade mineral óssea. Entre os artigos que investigaram diferentes subtipos histológicos, a maioria não reportou diferenças significativas entre os grupos. Considerando os estudos que avaliaram associação com tamanho da lesão, a maioria reportou uma associação positiva entre tamanho inicial e velocidade de redução. A maioria dos estudos que avaliou associação com a idade dos indivíduos concluiu que as técnicas parecem ser mais efetivas em pacientes mais jovens. Foram observados resultados satisfatórios das técnicas como tratamento preliminar na maioria dos estudos, além de serem reportadas como tratamento definitivo em três artigos. Portanto, as técnicas de marsupialização e descompressão demonstraram resultados satisfatórios na redução das lesões císticas como modalidades de tratamento preliminar, porém um longo período de acompanhamento é necessário para obtenção desses resultados.
Palavras-chave: Cistos maxilomandibulares. Descompressão. Marsupialização. Revisão
ABSTRACT
The purpose of this systematic review was to evaluate the effectiveness of surgical techniques of marsupialization and decompression on the reduction of cystic lesions of the jaws. Search strategies were developed for six electronic databases and three gray literature databases. Studies that evaluated the effectiveness of these therapies regarding reduction rates of cystic lesions, compared to measures before treatment, were considered eligible. A total of 1298 references were identified until May 2019, of which 27 before-after studies were included. Nine studies evaluated the marsupialization technique and eighteen decompression techniques. A high heterogeneity was observed between studies regarding outcome measures, such as percentage of reduction, Nakamura’s rating, lesion half-life, speed of shrinkage and increase on bone mineral density. Among included articles that investigated different histological subtypes, the majority reported no significant differences between groups. Considering the studies that evaluated association with lesion size, most reported a positive association between initial size and reduction speed. Most studies that investigated association with age of individuals concluded that the techniques appear to be more effective in younger patients. Satisfactory results of the techniques as preliminary treatment were observed in most studies, and were reported as definitive treatment in three articles. Therefore, marsupialization and decompression techniques presented overall satisfactory results on the reduction of cystic lesions as preliminary treatment modalities, although a long follow-up period is required to achieve these results.
LISTA DE FIGURAS
Do artigo em inglês:
Figure 1 - Flow diagram of literature search and selection criteria (adapted from Preferred
LISTA DE TABELAS
Do artigo em inglês:
Table 1 - Descriptive characteristics of included articles; marsupialization technique (n=9). 43
Table 2 - Descriptive characteristics of included articles; decompression technique (n=18).. 44
Table 3 - Risk of bias, assessed by the Joanna Briggs Institute Critical Appraisal Tools for
LISTA DE APÊNDICES
Do artigo em inglês:
Appendix 1 - Database search strategy. ... 47
Appendix 2 - Articles excluded and the reasons for exclusion (n=45). ... 49
Appendix 3 - Grading of Recommendations, Assessment, Development, and Evaluation
LISTA DE ANEXOS
Anexo A - Ata de apresentação do Trabalho de Conclusão de Curso ... 62
LISTA DE ABREVIATURAS E SIGLAS
DC - Císto dentígero
OKC - Ceratocisto odontogênico UA - Ameloblastoma unicístico
Do artigo em inglês:
CBCT - Cone-beam computed tomography CT - Computed tomography
DC - Dentigerous cyst
e.g. - Exempli grata (do latim)
GRADE - Grading of Recommendations Assessment, Development and Evaluation JBI - Joanna Briggs Institute
MA - Multicystic ameloblastoma mm2 - Square millimeter
OKC - Odontogenic keratocyst PR - Panoramic radiograph
PRISMA - Preferred Reporting Items for Systematic Reviews and Meta-Analysis PROSPERO - International Prospective Register of Systematic Reviews
RC - Radicular cyst RoB - Risk of bias
UA - Unicystic ameloblastoma SR - Systematic review
SUMÁRIO 1 INTRODUÇÃO ... 16 2 OBJETIVOS ... 18 2.1 Objetivo geral ... 18 2.2 Objetivos específicos ... 18 3 ARTIGO ... 19 4 CONCLUSÃO ... 58 REFERÊNCIAS ... 59
Anexo A – Ata da apresentação ... 62
1 INTRODUÇÃO
As lesões císticas são patologias muito prevalentes nos ossos maxilares (JOHNSON, BATSTONE e SAVAGE, 2013). Particularmente, as lesões de origem odontogênica são comumente assintomáticas, de crescimento lento e que podem envolver importantes estruturas anatômicas, tais como seios maxilares, nervo alveolar inferior e raízes dentárias (POGREL, 2005). Certas neoplasias benignas apresentam uma morfologia de aspecto cístico, como o ameloblastoma unicístico (UA), o que permite melhor resposta aos tratamentos conservadores frente a outras neoplasias (REICHART, PHILIPSEN e SONNER, 1995; ANTONOGLOU e SÁNDOR, 2014). Estas lesões podem apresentar grandes dimensões e aumentar consideravelmente o risco de danos às estruturas adjacentes relacionadas ao tratamento cirúrgico (GAO et al., 2018).
Embora os tratamentos cirúrgicos radicais sejam comumente considerados o tratamento de escolha para estas lesões (BATAINEH e AL QUDAH, 1998), graves complicações podem ocorrer após a cirurgia, incluindo deformidades faciais, fratura dos ossos maxilares, perdas dentárias e parestesia (SHEAR, 2002; WAKOLBINGER e BECK-MANNAGETTA, 2016). Além dos riscos relacionados à cirurgia, outros aspectos como idade do paciente, localização anatômica da lesão, tamanho e diagnóstico histopatológico também devem ser considerados no planejamento do tratamento, a fim de se obter melhores resultados e prognóstico (NAKAMURA et al., 2002b; WAKOLBINGER e BECK-MANNAGETTA, 2016). Nesse sentido, técnicas cirúrgicas conservadoras, como a marsupialização e a descompressão, podem ser opções adequadas, visto que são consideravelmente menos invasivas e possuem altas taxas de sucesso relatadas na literatura (KUBOTA et al., 2013; ALLON et al., 2015; LIZIO et al., 2018).
As técnicas de marsupialização e descompressão são frequentemente empregadas na cirurgia buco-maxilo-facial como tratamento preliminar para extensas lesões císticas, devido à relativa simplicidade e efetividade em comparação a outros tratamentos (CASTRO-NÚÑEZ, 2016). O procedimento de marsupialização consiste em incisar a parede cística e suturar o tecido patológico adjacente à mucosa oral, o que estabelece uma comunicação entre o lúmen cístico e a cavidade oral (POGREL e JORDAN, 2004). A técnica de descompressão, contudo, consiste na comunicação entre as cavidades cística e oral a partir da inserção de um dispositivo, como um dreno cirúrgico, na cavidade cística através da mucosa oral (MARKER et al., 1996). Ambas as técnicas visam a reduzir o tamanho das lesões a partir da liberação da pressão
17
intraluminal, o que favorece a neoformação gradual do tecido ósseo periférico à lesão (CATUNDA et al., 2013).
Ao reduzir parcial ou completamente o tamanho das lesões císticas, o risco de dano cirúrgico a importantes estruturas anatômicas pode ser minimizado e, em alguns casos, uma cirurgia secundária pode não ser necessária (POGREL e JORDAN, 2004; ULU et al., 2018). Esta redução pode ser avaliada através da porcentagem de redução, a qual compara medidas prévias e posteriores ao tratamento. Diante disso, Nakamura et al (2002b) propuseram um método para classificar o efeito da redução com base na resposta ao tratamento, sendo dividida em boa (redução da lesão acima de 80%), moderada (entre 50 e 80%) e ruim (abaixo de 50%).
Cabe ressaltar, no entanto, que as taxas de recorrência são frequentemente uma preocupação em relação a algumas lesões de aspecto cístico, como o OKC e o UA, tendo em vista seus comportamentos mais agressivos e invasivos (HENDRA et al., 2019). Logo, o uso de técnicas conservadoras como tratamento definitivo é controverso em relação a essas lesões e uma cirurgia secundária é frequentemente indicada (SLUSARENKO DA SILVA, STOELINGA e NACLÉRIO-HOMEM, 2019).
Os clínicos devem estar cientes da efetividade das técnicas de marsupialização e descompressão, a fim de fornecer recomendações baseadas em evidências sobre tratamentos com riscos reduzidos para seus pacientes (POGREL e JORDAN, 2004; WAKOLBINGER e BECK-MANNAGETTA, 2016). Revisões sistemáticas publicadas anteriormente focaram na efetividade dessas técnicas em relação às taxas de recorrência, sem, contudo, analisar as taxas de redução das lesões císticas (DE CASTRO et al., 2017; HENDRA et al., 2019; SLUSARENKO DA SILVA, STOELINGA e NACLÉRIO-HOMEM, 2019).
Sendo assim, esta é a primeira revisão sistemática a avaliar as taxas de redução como desfecho primário. Portanto, o objetivo deste estudo foi resumir e avaliar criticamente os dados da literatura sobre a efetividade das técnicas de marsupialização e descompressão na redução de lesões císticas dos maxilares.
2 OBJETIVOS
2.1 Objetivo geral
- Realizar uma síntese qualitativa e avaliação crítica da evidência disponível na literatura acerca da efetividade das técnicas cirúrgicas conservadoras de marsupialização e descompressão em relação à redução de lesões císticas dos maxilares, por meio de uma revisão sistemática da literatura.
2.2 Objetivos específicos
- Investigar possíveis diferenças nas taxas de redução considerando o diagnóstico histopatológico das lesões císticas;
- Explorar o efeito da idade e gênero dos participantes sobre a taxa de redução das lesões císticas;
- Verificar a influência do tamanho inicial das lesões sobre a taxa de redução das lesões císticas;
- Determinar possíveis diferenças na taxa de redução das lesões císticas considerando diferentes localizações anatômicas (maxila e mandíbula).
19
3 ARTIGO
Artigo formatado conforme as normas da revista Clinical Oral Investigations (acessadas em: 01/09/2019).
Title: Effectiveness of marsupialization and decompression on the reduction of cystic lesions
of the jaws: a systematic review.
Authors: Letícia Machado Berrettaa; Gilberto Melob; Fernanda Weber Mellob, Giuseppe Lizioc; Elena Riet Correa Riverod.
aDental School, Federal University of Santa Catarina - Florianópolis, Santa Catarina, Brazil. bPostgraduate program in Dentistry, Federal University of Santa Catarina - Florianópolis, Santa Catarina, Brazil.
cDepartment of Oral and Dental Sciences, University of Bologna, Bologna, Italy.
dDepartment of Pathology, Health Sciences Center, Federal University of Santa Catarina - Florianópolis, Santa Catarina, Brazil.
Corresponding author: Elena Riet Correa Rivero
Department of Pathology, Center of Health Sciences, Federal University of Santa Catarina, University Campus, Trindade, Florianópolis, 88.040-370, Santa Catarina, Brazil. Phone: +554837215068. E-mail: riet.elena@gmail.com
ABSTRACT
Objectives: The purpose of this systematic review was to evaluate the effectiveness of surgical
techniques of marsupialization and decompression on the reduction of cystic lesions of the jaws.
Materials and Methods: Search strategies were developed for six electronic databases and
three gray literature databases. Studies that evaluated the effectiveness of these therapies regarding reduction rates of cystic lesions, compared to measures before treatment, were considered eligible. Results: A total of 1298 references were identified, of which 27 before-after studies were included. Nine studies evaluated the marsupialization technique and eighteen decompression techniques. A high heterogeneity was observed between studies regarding outcome measures, such as percentage of reduction, Nakamura’s rating, lesion half-life, speed of shrinkage and increase on bone mineral density. Among included articles that investigated different histological subtypes, the majority reported no significant differences between groups. Considering the studies that evaluated association with lesion size, most reported a positive association between initial size and reduction speed. Most studies that investigated association with age of individuals concluded that the techniques appear to be more effective in younger patients. Satisfactory results of the techniques as preliminary treatment were observed in most studies, and were reported as a definitive treatment in three articles. In order to obtain satisfactory results, a long follow-up of the patients was observed. Conclusions: Marsupialization and decompression techniques presented overall satisfactory results on the reduction of cystic lesions as preliminary treatment modalities, although a long follow-up period is required to achieve these results.
Clinical relevance: The knowledge provided by this SR may be useful for a better clinical
decision on the management of patients with cystic lesions of the jaws. These data might help clinicians to perform less invasive treatments to their patients.
21
INTRODUCTION
Cystic lesions are one of the most prevalent pathologies of the jaws [1]. Those with odontogenic origin are commonly asymptomatic, slow-growing, and with possible involvement of important anatomical structures such as maxillary sinuses, inferior alveolar nerve, and dental root [2]. Several cystic lesions such as odontogenic keratocysts (OKC), dentigerous cysts (DC), and unicystic ameloblastomas (UA) can present large dimensions and thus the risk of damage to adjacent structures related to surgical treatment is considerably increased [3].
Although radical surgical treatments (eg. radical resection) are commonly considered the treatment of choice [4], serious complications may occur following surgery, including facial deformities, maxillary bone fractures, dental losses, and paresthesia [5, 6]. Notwithstanding surgery-related risks, other features such as patients' age, lesion anatomical location, size, and histological diagnosis should also be considered in treatment planning to achieve better treatment responses and prognosis [5, 7]. In this regard, conservative surgical techniques such as marsupialization and decompression might be suitable options since these surgical procedures are considerably less invasive and overall high success-rates have been reported in the literature [8-10].
Both marsupialization and decompression techniques are often used in the field of maxillofacial surgery to manage large jaw cysts lesions due to its relatively simplicity and effectiveness compared to other treatments [11]. Marsupialization technique consists of an incision directly through the cystic wall and suture the pathological tissue adjacent to oral mucosa, which establish a communication between the cystic lumen and oral cavity [12]. Meanwhile, the decompression technique consists of suturing a device, such as a surgical stent, across the cystic cavity and the oral mucosa, which allows constant drainage and bone growth [13]. Both techniques aim to reduce the cystic lesions by releasing the intraluminal pressure, which favors gradual bone tissue neoformation [14].
By diminishing cystic lesions' size, either partially or completely, the risk of surgical damage to important anatomical structures can be minimized or, in some cases, a secondary surgery might not be required [12, 15]. It must be highlighted, however, that recurrence rates are frequently a major concern regarding several cystic lesions, such as the OKC and UA [16]. Therefore, the use of conservative techniques as definitive treatments is controversial with regards to these lesions and a secondary surgery is often indicated [17].
Clinicians must be aware of the effectiveness of these conservative treatments in order to provide evidence-based treatment recommendations with minimal risks to their patients [12]. However, previously published systematic reviews (SR) have focused on recurrence rates rather than reduction of cystic jaw lesions per se [16-18]. To the best of authors' knowledge, this is the first SR to assess reduction rates as primary outcome. Therefore, the purpose of this SR was to summarize and critically appraise data from the literature concerning the effectiveness of marsupialization or decompression techniques in the reduction of cystic lesions of the jaws.
METHODS
This SR was conducted according to the guidelines of the Preferred Reporting Items for Systematic Reviews and Meta-Analysis checklist (PRISMA) [19]. The study protocol was registered at the International Prospective Register of Systematic Reviews (PROSPERO) [20] under the registration number CRD42019116099.
Eligibility criteria
Inclusion criteria
The acronym PICOS (Population, Intervention, Comparison, Outcomes, Studies) was used to elaborate the focused question of this study, in which: P) humans with cystic lesions of the jaws; I) marsupialization and/or decompression; C) other therapies or no comparison group (before-after design); O) cystic lesion reduction; and S) randomized clinical trials, non-randomized clinical trials, and case-series with at least 10 lesions treated with marsupialization and/or decompression. Only articles published in the Latin Roman alphabet were considered. No time restriction was applied.
Exclusion criteria
The following exclusion criteria were applied: 1) studies that did not investigate marsupialization and/or decompression techniques; 2) studies that did not report outcomes related to lesion reduction; 3) case-reports, reviews, letters, conference abstracts, personal opinions, laboratory research, and case-series with less than 10 cases of marsupialization and/or decompression; 4) studies not published in the Latin Roman alphabet; 5) full-text not available.
23
Search strategies were elaborated and adapted for six main electronic databases: EMBASE, Latin American and Caribbean Health Sciences (LILACS), PubMed, Scopus, The Cochrane Library, and Web of Science. In addition, a grey literature search was performed on Google Scholar, OpenGrey, and ProQuest. All searches were performed on May 8, 2019. Detailed search strategies are reported in Appendix 1. The reference lists of included articles were also hand-searched for potentially eligible studies, as recommended by Greenhalgh and Peackock (2005) [21]. Reference management and duplicates removal were performed using a reference manager software (Endnote X7, Thomson Reuters, Philadelphia, USA).
Study selection
The selection process was performed in two phases using an online software (Rayyan, Qatar Computing Research Institute). In phase-1, two blinded reviewers (L.M.B. and G.M.) independently screened titles and abstracts of all references in order to identify eligible studies. In phase-2, the same two reviewers performed a full-text reading of selected studies. Any disagreements regarding eligibility criteria were resolved by a consensus discussion and, if necessary, a third reviewer (F.W.M.) was consulted to make the final decision.
Data collection process and data items
Two reviewers (L.M.B. and G.M.) independently collected pertinent data from included articles, and information was then cross-checked to assure integrity of contents. Gathered data encompassed studies features (authors, year of publication, country, and study design), population characteristics (sample size, study groups, mean age, lesion location and diagnosis) and outcome characteristics (surgical approach, assessment method, intervention duration, time of follow-up and main findings).
Risk of bias in individual studies
The risk of bias (RoB) of the included articles was assessed separately by two reviewers (L.M.B and G.M.) using the Joanna Briggs Institute Critical Appraisal Checklist for Quasi-Experimental Studies (non-randomized experimental studies) [22]. In case of disagreements, a third reviewer (F.W.M.) was involved. Decisions about scoring system and cut-off points were agreed upon by all reviewers before critical appraisal commences, as advised by the JBI reviewers’ manual [23]. Possible answers were “yes”, “unclear”, “no” or “not applicable”. The studies were categorized as “low” when the study reached more than 70% score “yes”;
“moderate” when reaching 50% to 69% score “yes”; and “high” when reaching up to 49% score “yes”. The software RevMan 5.3 (Review Manager 5.3, The Nordic Cochrane Centre, Copenhagen, Denmark) was used to generate figures.
Summary measures
Reduction rates or equivalent data (e.g. percentage of reduction, speed of shrinkage) related to marsupialization and/or decompression of cystic lesions of the jaws were considered as the primary outcomes of this SR. If data were available, recurrence rates and other complications were considered as secondary outcomes. Any summary measures for continuous or categorical data were considered. Statistical significance was set at α = 5%.
Synthesis of results
A qualitative synthesis of results was performed. Statistical pooling of data using meta-analysis was planned if included studies were considered sufficiently homogeneous in regards to study design, follow-up times, cystic reduction assessment criteria, and outcome summary measures.
Risk of bias across studies
Methodological heterogeneity was assessed by comparing the variability in the RoB and study design of included studies. Clinical heterogeneity was evaluated by investigating variability related to studies' population (e.g. age and sex) and outcomes (e.g. histological diagnosis of cystic lesions and lesion initial size).
Additional analyses
An assessment of certainty in cumulative evidence was performed using the "Grading of Recommendations Assessment, Development and Evaluation" (GRADE) criteria [24] and a summary of findings table was produced using an online software (GRADEpro GDT, McMaster University).
RESULTS
25
After duplicates had been removed, a total of 1298 studies remained for title and abstracts screening (phase-1), of which 72 were considered eligible to be fully assessed. After full-text reading (phase-2), 27 studies were finally included for the qualitative synthesis. An overview of the selection process is shown in Figure 1.
Study characteristics
All included articles were before-after studies. A total of 861 individuals were enrolled, of which 40% were women. Age of participants ranged from 6 [25] to 79 [26] years old and the most prevalent age range was 30 to 40 years old. A total of 885 cystic lesions were found, ranging from 10 [12] to 78 [27] lesions per study. The most prevalent lesions investigated across studies were OKC (n=355), followed by DC (n=222), UA (n=154), and RC (n=78).
Regarding surgical approach, 18 studies assessed the decompression technique [3, 8, 9, 15, 26, 28-40], while marsupialization was investigated in 9 studies [7, 10, 12, 25, 27, 41-44]. Considering lesion anatomical location, both maxillary and mandibular lesions were investigated in 12 articles [3, 7, 8, 12, 26-28, 30, 33, 35-37], while in 14 studies only mandibular lesions were assessed [9, 10, 15, 25, 29, 31, 34, 38-44]. In addition, information concerning lesion anatomical location was not available in a sole study [32]. With regards to lesion reduction assessment method, 18 studies used panoramic radiographs (PR) [7-10, 12, 25, 27, 28, 30-33, 36-38, 41-44] and 12 studies used computed tomography (CT) [3, 15, 26, 29, 31, 34-36, 39, 40, 42, 43], of which 3 reported the use of cone-beam computed tomography (CBCT) [3, 34, 35]. In addition, the criteria proposed by Nakamura et al. (2002b) [7] for rating lesion reduction based on treatment response (good, moderate, and poor) was used in 8 studies [7, 8, 26-28, 31, 33, 43].
Intervention duration ranged from 2.7 months [32] to approximately 25 months [25]. Time of follow-up after intervention ranged from at least 6 months [37] to 6.6 years [7]. Eleven articles reported recurrence rates during post-intervention follow-up periods [7, 25, 27, 28, 30, 31, 33, 36, 40, 43] and no recurrences were reported in 3 of these studies [30, 31, 36]. Information regarding time of follow-up was not available in 9 articles [3, 10, 26, 29, 32, 34, 38, 39, 42]. Furthermore, three studies reported marsupialization [12] and decompression [9, 15] techniques as the definitive treatment for cystic lesions.
Marsupialization technique
Detailed characteristics regarding the marsupialization technique can be found in Table 1. From studies that reported percentage of reduction regarding cystic lesions, one study reported a mean reduction higher than 80% [7], two studies between 50% and 79% [25, 27], while in one study the percentage of reduction was lower than 50% [43]. Considering studies that reported Nakamura’s rating [7, 27, 43], the majority of lesions (more than 50%) presented a good response of reduction in only one study [7]. Furthermore, a sole study reported completely resolution for all lesions (OKC) after marsupialization [12].
Regarding cystic lesions' half-life (characterized by the time required for a 50% lesion reduction), Shudou et al. (2012) [42] reported a mean half-life of approximately 8 months for OKC, while Kubota et al. (2013) [10] reported a mean half-life of 12 months for DC, 9 months for OKC, and 6 months for RC. Furthermore, regarding speed of shrinkage of cystic lesions, Miyawaki et al. (1999) [41] reported a mean absolute speed of shrinkage of 224.0±352.0mm2/month for DC, while Kubota et al. (2013) [10] reported that RC presented a significantly higher mean relative speed of shrinkage compared to other lesions, although no significant differences were found between DC and OKC.
Regarding individuals' age, two studies reported that marsupialization was less effective in older participants [7, 44], whilst one reported no differences in duration of procedure considering patients' age [43]. With regards to participants' sex, a sole study reported no differences considering increase in bone density [44]. Considering lesion initial size, two studies reported faster speed of shrinkage for larger lesions [10, 42], and one study reported faster bone regeneration in larger lesions [44]. Moreover, two studies investigating differences across histological diagnoses reported a better treatment response in UA compared to multicystic ameloblastoma [25, 27] and one study reported a higher speed of shrinkage for RC compared to OKC [10].
Decompression technique
Detailed characteristics are available in Table 2. Percentage of reduction was the most common method to evaluate cystic lesions reduction with decompression technique, assessed by 12 studies and ranging from approximately 48.2% [29] to 87.5% [9]. Three studies reported a percentage of reduction higher than 80% [8, 9, 31], seven studies between 50% and 79% [26,
27
28, 30, 33, 35, 36, 40], while in two studies the percentage of reduction was lower than 50% [29, 34]. In addition, three studies reported percentage of reduction according to histological diagnosis [32, 33, 38]. The reduction of DC ranged from 39.4% [38] to 62.6% [33], OKC from 33.6% [38] to 78.9% [32], RC from 28.0% [33] from 79.6% [32], and UA from 36.7% [38] to 67.2% [32]. Furthermore, two studies reported completely resolution for all lesions (DC, OKC, and RC) after decompression [9, 15].
From Nakamura’s rating evaluation, three studies reported a majority of good response on lesion reduction [8, 28, 31], while 2 studies reported moderate response on the reduction [26, 33]. No study reported a majority of poor response on cystic lesions reduction. Regarding speed of shrinkage after decompression, Oliveros-Lopez et al. (2017) reported a mean of 31.7±19.4mm2/month for DC and 13.7±6.4mm2/month for RC [37], whilst Gao et al. (2014) reported no significant differences among OKC, RC, and UA [32]. As exception, Gao et al. (2018) evaluated increase in bone mineral density based on CBCT scans after decompression, of which no significant differences were observed among DC, OKC, and UA.
Regarding individual’s age, three studies reported higher reduction rates in younger individuals [28, 38, 39], while one study reported higher monthly reduction rates in older age [37]. Still, one study reported no differences in reduction rates regarding individual’s age [35]. Among studies that evaluated lesion anatomical location, two studies reported significant higher reduction rates in mandibular cysts [33, 37], whilst 2 studies reported no difference between maxilla and mandible [8, 28]. No study reported a significant higher reduction rate considering lesions located in the maxilla. Furthermore, with regards to cysts' size, initial volume was significantly associated with lesion reduction rates in 2 studies [35, 39]. One study reported faster reduction in smaller cysts [28], whilst 3 studies reported higher reduction rates in larger lesions [26, 37, 38].
Among histological diagnosis groups of DC, OKC, RC and UA, four studies reported no significant differences in reduction rates [26, 28, 29, 39], two study reported no significant differences in speed of shrinkage [32, 38], and one study reported no differences in increase of bone mineral density [3]. Nevertheless, one study reported faster increase in bone density in RC than in OKC and UA [32], whilst one study reported faster speed of shrinkage for DC compared to RC [37].
With regards to marsupialization, mean intervention duration was 15.6 months. A total of 107 lesions were graded according to Nakamura’s rating, of which 42(39.2%) presented a good treatment response, 38(35.5%) moderate, and 27(25.3%) poor response. Percentage of reduction was higher than 70% in most studies, although a poor response was found in a single study (36.8%) [43]. In addition, mean absolute speed of reduction was only reported for DC (224±352mm2/month) [41]. No differences in relative speed of reduction were found for DC and OKC, whilst higher speeds were observed for RC. Lesion half-life was reported in 2 studies, in which a 50% of reduction was observed within approximately 12 months for DC, 8.5 months for OKC and 6 months for RC [10, 42].
Furthermore, with regards to the decompression technique, the mean intervention duration across studies was approximately 9.9 months. From a total of 188 lesions graded according to Nakamura’s rating, 89 presented a good response, 70 moderate response, and 29 a poor response. Most studies reported moderate percentage of reduction, while two studies reported poor reduction rates [29, 34]. Regarding speed of shrinkage, DC responded better than RC in one study [37], whilst one study reported no significant differences among OKC, RC, and UA [32]. Considering increase in bone mineral density, one study reported faster increase in RC than in OKC and UA [32], while one study reported no significant differences regarding histological groups [3].
Risk of bias within studies
Overall, two studies were judged as with high risk [28, 44], twenty-three as with moderate risk [3, 7, 8, 10, 12, 15, 25-27, 29-39, 41-43], and 2 studies as with low risk of bias [9, 40]. Several domains were judged with high RoB majorly due to: 1) the absence of control groups; 2) substantial variability regarding participants included in comparisons involving participants' age/gender, lesions' histological diagnosis, initial size, and anatomical location; and 3) lack of detailed information regarding assessment method for lesion reduction. More information concerning RoB evaluation is provided in Table 3.
Risk of bias across studies
The studies were considered heterogeneous in their methods regarding differences in sample size, participants' age, intervention duration, time of follow-up and lesion reduction assessment method. Moreover, different surgical approaches were described, such as
29
marsupialization and decompression alone or as followed by a secondary and definitive treatment. Furthermore, different outcome measures were reported, such as percentage of reduction, Nakamura’s rating, lesions’ half-life, speed of shrinkage and increase on bone mineral density,
Additional analyses
The certainty in cumulative evidence assessed using GRADE criteria was judged as very low. This indicate that true effect is likely to be substantially different from the estimated effect. Domains of concern were related to: 1) risk of bias, according to overall judgments of the JBI critical appraisal tools; 2) inconsistency, due to heterogeneous lesion reduction assessment methods, variability in follow-up times, and considerably different reduction rates observed; 3) indirectness, related to limited validity of results' extrapolation, which as attributable to possible differences in effect estimate not addressed in the overall effect measure provided; 4) imprecision, due to poorly reported variability measures and moderate to considerable imprecise data. Further information is provided in Appendix 3.
DISCUSSION
The available evidence concerning the effectiveness of marsupialization and decompression techniques in the reduction of jaw cystic lesions was assessed in this SR. Overall satisfactory treatment responses and low complication rates were found across included studies. Moreover, a reduced risk of damage to adjacent structures were often considered an advantage of these conservative techniques [45], thus clinicians must be aware of its effectiveness in order to improve treatment planning and provide evidence-based recommendations for their patients. Data from included articles were considerably heterogeneous due to different outcome measures, such as percentage of reduction, Nakamura’s rating, lesions’ half-life, speed of shrinkage, and increase in bone mineral density.
The assessment of lesions' reduction was hindered due to multiple outcome measures, nonetheless, it was found that long follow-up spans, both during and post-intervention, were usually necessary for the treatment to be considered successful. In this context, some concerns were raised regarding patients' adherence to long-term interventions, which could result in high rates of patients’ dropout [46]. The viability of carrying out long-term treatments should be discussed in order to obtain satisfactory results, once success of conservative techniques usually
depends on patients’ cooperation [47]. Therefore, the need to previously inform patients about time spans required for satisfactory reduction rates with conservative treatments is highlighted.
Bone regeneration of residual cystic cavities has complex biological foundations, which depends on bone remodeling metabolism and nature of involved tissues [48]. In this regard, several included studies reported that individuals’ age might affect treatment response and that younger patients usually presents more satisfactory reduction rates with conservative treatments [7, 28, 38, 39, 44]. This finding might be partially explained due to greater potential of bone regeneration of younger individuals when compared to older populations [48]. It should be mentioned that since the craniofacial skeleton is still in development stage in pediatric populations, conservative surgical protocols should be considered in order to avoid damage to developing teeth or other facial structures [8]. Nonetheless, although available evidence was somewhat sparse considering age's influence on treatment response, this topic should be further explored since possible benefits of this conservative technique for younger populations cannot be ruled out.
Regarding lesion initial size, most included studies reported higher reduction rates and faster bone regeneration for larger lesions [10, 26, 37, 38, 42, 44], while a single study reported faster reduction in smaller lesions [28]. Furthermore, it should be mentioned that treatment response considering lesion location was not extensively investigated and results found were somewhat conflicting, as two studies did not find significant differences [8, 28], whilst other two reported a slightly better treatment response with regards to mandibular cysts [33, 37]. Possible mechanisms involved regarding effects of both initial size and lesion location on treatment response were not widely discussed in current literature, thus further studies are recommended to investigate these topics
Considering the different histological diagnoses of cystic lesions, most studies investigating DC, OKC, RC and UA reported no significant differences on the reduction rates across these lesions [3, 26, 28, 29, 38, 39]. Nonetheless, a better treatment response of RC compared to other lesions was observed in 2 studies [10, 32]. Although there is no consensus in current literature, several studies reported that RC may present lower immunoexpression of matrix metalloproteinase-2, which is proposed to be related to lesions' growth and progression, thus potentially affecting treatment response with conservative techniques [49].
31
Furthermore, two studies reported a better treatment response on reduction of UA compared to multicystic ameloblastoma (MA), which corroborate with extensive studies in literature; it is proposed that UA could present good treatment responses to conservative treatments, while radical procedures are often recommended for the management MA [50, 51]. Due to the morphology of multicystic ameloblastoma, a satisfactory reduction after marsupialization was not expected. This may be explained because UA lesions are well delimited by a fibrous capsule, while MA lesions tend to aggressively invade cancellous bone, which could also result in higher recurrence rates [52]. Another hypothesis was proposed by Zhang et al. (2018), which found that patients with UA that had higher expression of Interleukin-1a (IL-1a) - an inflammatory cytokine involved in tumor growth - presented better responses to marsupialization, thus proposing that UA might share similar biological behavior as OKC [43].
In this regard, several studies investigating OKC in particular reported that the expression of IL-1a and Ki-67 (a cell proliferation marker) is inhibited following marsupialization treatment [49]. These findings might explain possible mechanisms by which the release of intraluminal pressure can result in reduction of cystic lesions with aggressive behavior [53, 54]. Nonetheless, although conservative techniques may promote the downregulation of several cell signaling pathways related to cystic growth and development, studies with long-term follow-up spans are necessary to investigate this topic as it may take years before evident lesion recurrence to occur [52].
Although recurrence rates are not the only factor to be considered in treatment planning, caution should be exercised whenever conservative techniques are used for lesions with high recurrence potential. From four studies that reported marsupialization or decompression techniques as the definitive treatment for most lesions (including DC, OKC, and RC) [8, 9, 12, 15], only two clearly reported no lesion recurrences [8, 15]. Most published SR on this topic have assessed syndromic or non-syndromic OKC, while only one SR was found regarding UA. In this regard, radical resection presented the lowest recurrence rates in six SR [51, 55-59], while decompression followed by enucleation presented lower rates of recurrence in one SR [18]. In addition, from SR investigating marsupialization in particular [17, 60], one reported that marsupialization and delayed enucleation presented lowest rates of recurrence compared to enucleation alone [17], while the other proposed that cystectomy (with or without adjuvant therapy) is associated with fewer recurrences [60]. Notwithstanding these findings, no clear
recommendation regarding the best treatment option based solely on recurrence rates was provided in most of these SR, which reported that available evidence was severely affected by lack of well-designed long-term studies.
Other complications and surgery related risks considering conventional therapies, especially for larger lesions, had been widely discussed in the literature [5, 6]. In regards to conservative techniques in particular, although complication rates were not the primary outcome of this SR, it must be mentioned that lesion infection may be facilitated due to communication between cystic and oral cavities in the marsupialization technique [37], whilst the possibility of breaking and need for adjustments of the stent in the decompression technique should also be considered [47]. Therefore, the need for patients' collaboration considering conservative treatments for cystic jaw lesions is highlighted.
Limitations
All included studies were of before-after design, which provides limited evidence with regards to effectiveness comparison across treatment options currently available [61]. In addition, articles were considerably heterogeneous regarding sample size, participants' age, intervention duration, follow-up time, lesion reduction assessment method, and outcome measures. Per reasons described, a statistical pooling of data using meta-analysis was not considered appropriate. Thus, results must be interpreted with caution and further controlled studies comparing treatment modalities with regards to lesion reduction and bone formation are recommended.
CONCLUSION
Marsupialization and decompression techniques presented overall satisfactory reduction rates as preliminary treatment modalities for cystic lesions of the jaws, being considered as the definitive treatment for most lesions in several studies. Nonetheless, considerably long follow-up spans were usually required for satisfactory treatment responses to be achieved. Due to lack of well-designed controlled trials, clinicians should interpret these findings with caution.
33
Acknowledgments
FWM (grant number 88887.200724/2018-00) and GM (grant number 88887.200723/2018-00) are supported by the Fundação de Amparo à Pesquisa e Invovação do Estado de Santa Catarina (FAPESC).
Compliance with ethical standards
Conflict of interest: The authors declare that they have no conflict of interest.
Ethical approval: This work does not contain any studies with human participants or animals
performed by any of the authors.
References
1. Johnson NR, Gannon OM, Savage NW and Batstone MD (2014) Frequency of odontogenic cysts and tumors: a systematic review. J Investig Clin Dent 5:9-14.
2. Pogrel MA (2005) Treatment of Keratocysts: The Case for Decompression and Marsupialization. J Oral Maxillofac Surg 63:1667-1673.
3. Gao L, Ren WH, Li SM, Zheng JJ, Xue LF, Xu YX, Wang QB, Song JZ, Dou ZC, Zhou MZ, Xiao WL and Zhi KQ (2018) CBCT-based bone quality assessment in decompression of large odontogenic cystic lesions. Oral Radiol 34:251-256.
4. Bataineh AB and Al Qudah M (1998) Treatment of mandibular odontogenic keratocysts. Oral Surg Oral Med Oral Pathol Oral Radiol 86:42-47.
5. Wakolbinger R and Beck-Mannagetta J (2016) Long-term results after treatment of extensive odontogenic cysts of the jaws: a review. Clin Oral Investig 20:15–22.
6. Shear M (2002) The aggressive nature of the odontogenic keratocyst: is it a benign cystic neoplasm? Part 1. Clinical and early experimental evidence of aggressive behaviour. Oral Oncol 38:219-226.
7. Nakamura N, Mitsuyasu T, Mitsuyasu Y, Taketomi T, Higuchi Y and Ohishi M (2002b) Marsupialization for odontogenic keratocysts: long-term follow-up analysis of the effects and changes in growth characteristics. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 94:543-553.
8. Allon DM, Allon I, Anavi Y, Kaplan I and Chaushu G (2015) Decompression as a Treatment of Odontogenic Cystic Lesions in Children. J Oral Maxillofac Surg 73:649-654.
9. Lizio G, Ferraioli L, Melini M and Marchetti C (2018) Long-term investigation of decompression as a definitive treatment for mandibular cysts associated with impacted third molars. J Am Dent Assoc:1-7.
10. Kubota Y, Imajo I, Itonaga R and Takenoshita Y (2013) Effects of the patient's age and the size of the primary lesion on the speed of shrinkage after marsupialisation of keratocystic
35
odontogenic tumours, dentigerous cysts, and radicular cysts. Br J Oral Maxillofac Surg 51:358-362.
11. Castro-Núñez J (2016) Decompression of Odontogenic Cystic Lesions: Past, Present, and Future. J Oral Maxillofac Surg 74:104.e1-9.
12. Pogrel MA and Jordan RCK (2004) Marsupialization as a Definitive Treatment for the Odontogenic Keratocyst. J Oral Maxillofac Surg 62:651-655.
13. Marker P, Brondum N, P. CP and Bastian HL (1996) Treatment of large odontogenic keratocysts by decompression and later cystectomy: a long-term follow-up and a histologic study of 23 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 82:122-131.
14. Catunda IS, Catunda RB, Vasconcelos BC and de Oliveira HF (2013) Decompression device for cavitary bone lesions using Luer syringe. J Oral Maxillofac Surg 71:723-725.
15. Ulu M, Kelebek S, Kalabalik F and Akcay H (2018) Correlation between decompression period and initial volume of large cystic lesions of the mandible: a retrospective CBCT study. Cukurova Medical Journal 43:254-260.
16. Hendra FN, Natsir Kalla DS, Van Cann EM, de Vet HCW, Helder MN and Forouzanfar T (2019) Radical vs conservative treatment of intraosseous ameloblastoma: Systematic review and meta-analysis. Oral Dis 00:1-14.
17. Slusarenko da Silva Y, Stoelinga PJW and Naclério-Homem MDG (2019) Recurrence of nonsyndromic odontogenic keratocyst after marsupialization and delayed enucleation vs. enucleation alone: a systematic review and meta-analysis. Oral Maxillofac Surg 23:1-11.
18. De Castro MS, Caixeta CA, De Carli ML, Ribeiro Júnior NV, Miyazawa M, Pereira AAC, Sperandio FF and Hanemann JAC (2017) Conservative surgical treatments for nonsyndromic odontogenic keratocysts: a systematic review and meta-analysis. Clin Oral Investig 22:2089-2101.
19. Moher D, Liberati A, Tetzlaff J, Altman DG and Group P (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med 151:264-9, W64.
20. Booth A, Clarke M, Ghersi D, Moher D, Petticrew M and Stewart L (2011) An international registry of systematic-review protocols. Lancet 377:108-9.
21. Greenhalgh T and Peacock R (2005) Effectiveness and efficiency of search methods in systematic reviews of complex evidence: audit of primary sources. BMJ 331:1064-5.
22. Tufanaru C, Munn Z, Aromatis E, Campbell J and Hopp L (2017) Chapter 3: Systematic reviews of effectiveness. In: Aromatis E and Munn Z (eds) Joanna Briggs Institute Reviewer's Manual. The Joanna Briggs Institute, Adelaide
23. Joanna Briggs Institute (2014) Joanna Briggs institute reviewers’ manual: 2014 edition. The Joanna Briggs Institute, Adelaide
24. Guyatt GH, Oxman AD, Vist GE, Kunz R, Falck-Ytter Y, Alonso-Coello P and Schunemann HJ (2008) GRADE: an emerging consensus on rating quality of evidence and strength of recommendations. BMJ 336:924-6.
25. Yang Z, Liang Q, Yang L, Zheng GS, Zhang SE, Lao XM, Liang YJ and Liao GQ (2018) Marsupialization of mandibular cystic ameloblastoma: Retrospective study of 7 years. Head Neck Surg:2171-2178.
26. Jeong HG, Hwang JJ, Lee SH and Nam W (2017) Effect of decompression for patients with various jaw cysts based on a three-dimensional computed tomography analysis. Oral Surg Oral Med Oral Pathol Oral Radiol 123:445-452.
27. Nakamura N, Higuchi Y, Mitsuyasu T, Sandra F and Ohishi M (2002a) Comparison of long-term results between different approaches to ameloblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 93:13-20.
28. Anavi Y, Gal G, Miron H, Calderon S and Allon DM (2011) Decompression of odontogenic cystic lesions: clinical long-term study of 73 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 112:164-169.
29. Asutay F, Atalay Y, Turamanlar O, Horata E and Burdurlu MC (2016) Three-Dimensional Volumetric Assessment of the Effect of Decompression on Large Mandibular Odontogenic Cystic Lesions. J Oral Maxillofac Surg 74:1159-1166.
37
30. August M, Faquin WC, Troulis MJ and Kaban LB (2003) Dedifferentiation of odontogenic keratocyst epithelium after cyst decompression. J Oral Maxillofac Surg 61:678-683.
31. Enislidis G, Fock N, Sulzbacher I and Ewers R (2004) Conservative treatment of large cystic lesions of the mandible: A prospective study of the effect of decompression. Br J Oral Maxillofac Surg 42:546-550.
32. Gao L, Wang XL, Li SM, Liu CY, Chen C, Li JW, Yan XJ, Zhang J, Ren WH and Zhi KQ (2014) Decompression as a Treatment for Odontogenic Cystic Lesions of the Jaw. J Oral Maxillofac Surg 72:327-333.
33. Lee ST, Kim SG, Moon SY, Oh JS, You JS and Kim JS (2017) The effect of decompression as treatment of the cysts in the jaws: retrospective analysis. J Korean Assoc Oral Maxillofac Surg 43:83-87.
34. Lizio G, Sterrantino AF, Ragazzini S and Marchetti C (2013) Volume reduction of cystic lesions after surgical decompression: a computerised three-dimensional computed tomographic evaluation. Clin Oral Invest 17:1701-1708.
35. Lizio G, Tomaselli L, Landini L and Marchetti C (2017) Dentigerous cysts associated with impacted third molars in adults after decompression: a prospective survey of reduction in volume using computerised analysis of cone-beam computed tomographic images. Br J Oral Maxillofac Surg 55:691-696.
36. Oh JS, You JS and Kim SG (2018) Clinical and histomorphometric evaluation of decompression followed by enucleation in the treatment of odontogenic keratocyst. J Dent Science XX:1-5.
37. Oliveros-Lopez L, Fernandez-Olavarria A, Torres-Lagares D, Serrera-Figallo MA, Castillo-Oyagüe R, Segura-Egea JJ and Gutierrez-Perez JL (2017) Reduction rate by decompression as a treatment of odontogenic cysts. Med Oral Patol Oral Cir Bucal 22:643-650.
38. Park HS, Song IS, Seo BM, Lee JH and Kim MJ (2014) The effectiveness of decompression for patients with dentigerous cysts, keratocystic odontogenic tumors, and unicystic ameloblastoma. J Korean Assoc Oral Maxillofac Surg 40:260-5.
39. Song IS, Park HS, Seo BM, Lee JH and Kim MJ (2015) Effect of decompression on cystic lesions of the mandible: 3-dimensional volumetric analysis. Br J Oral Maxillofac Surg 53:841-848.
40. Park JH, Kwak EJ, You KS, Jung YS and Jung HD (2019) Volume change pattern of decompression of mandibular odontogenic keratocyst. Maxillofac Plast Reconstr Surg 41:2.
41. Miyawaki S, Hyomoto M, Tsubouchi J, Kirita T and Sugimura M (1999) Eruption speed and rate of angulation change of a cyst-associated mandibular second premolar after marsupialization of a dentigerous cyst. Am J Orthod Dentofacial Orthop 116:578-584.
42. Shudou H, Sasaki M, Yamashiro T, Tsunomachi S, Takenoshita Y, Kubota Y, Ninomiya T, Kawazu T and Mori Y (2012) Marsupialisation for keratocystic odontogenic tumours in the mandible: longitudinal image analysis of tumour size using 3D visualised CT scans. Int J Oral Maxillofac Surg 41:290-296.
43. Zhang X, Liu L, Yang X, Wang L, Zhang C and Hu Y (2018) Expression of TP53 and IL-1a in unicystic ameloblastoma predicts the efficacy of marsupialization treatment. Medicine (United States) 97.
44. Zhao Y, Liu B, Han QB, Wang SP and Wang YN (2011) Changes in bone density and cyst volume after marsupialization of mandibular odontogenic keratocysts (Keratocystic Odontogenic Tumors). J Oral Maxillofac Surg 69:1361-1366.
45. Eyre J and Zakrzewska JM (1985) The conservative management of large odontogenic keratocysts. Br J Oral Maxillofac Surg 23:195-203.
46. Maurette PE, Jorge J and Moraes M (2006) Conservative Treatment Protocol of Odontogenic Keratocyst: A Preliminary Study. J Oral Maxillofac Surg 64:379-383.
47. Rahpeyma A and Khajehahmadi S (2016) Marsupialization for Treatment of Jaw Cysts: Indications and Limitations. J Int Oral Health 8:158-162.
48. Rubio ED and Mombrú CM (2015) Spontaneous Bone Healing after Cysts Enucleation without Bone Grafting Materials: A Randomized Clinical Study. Craniomaxillofac Trauma Reconstr 8:14-22.
39
49. Khalifa GA, Shokier HM and Abo-Hager EA (2010) Evaluation of neoplastic nature of keratocystic odontogenic tumor versus ameloblastoma. J Egypt Natl Canc Inst 22:61-72.
50. Reichart PA, Philipsen HP and Sonner S (1995) Ameloblastoma: Biological profile of 3677 cases. Oral Oncol, Eur J Cancer 31B:86-99.
51. Antonoglou GN and Sándor GK (2014) Recurrence rates of intraosseous ameloblastomas of the jaws: A systematic review of conservative versus aggressive treatment approaches and meta-analysis of non-randomized studies. J Oral Maxillofac Surg 43:149-157.
52. Gardner DG (1984) A Pathologist's Approach to the Treatment of Ameloblastom. J Oral Maxillofac Surg 42:161-165.
53. Ninomiya T, Kubota Y, Koji T and Shirasuna K (2002) Marsupialization inhibits interleukin-1aexpression and epithelial cell proliferationin odontogenic keratocysts. J Oral Pahol Med 31:526-533.
54. Suyama Y, Kubota Y, Yamashiro T, Ninomiya T, Koji T and Shirasuna K (2009) Expression of keratinocyte growth factor and its receptorin odontogenic keratocysts. J Oral Pathol Med 38:476-480.
55. Al-Moraissi EA, Dahan AA, Alwadeai MS, Oginni FO, Al-Jamali JM, Alkhutari AS, Al-Tairi NH, Almaweri AA and Al-Sanabani JS (2017) What surgical treatment has the lowest recurrence rate following the management of keratocystic odontogenic tumor?: A large systematic review and meta-analysis. J Craniomaxillofac Surg 45:131-144.
56. Blanas N, Freund B, Schwartz M and Furst IM (2000) Systematic review of the treatment and prognosis of the odontogenic keratocyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 90:553-558.
57. Johnson NR, Batstone MD and Savage NW (2013) Management and recurrence of keratocystic odontogenic tumor: a systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol 116.
58. Kaczmarzyk T, Mojsa I and Stypulkowska J (2012) A systematic review of the recurrence rate for keratocystic odontogenic tumour in relation to treatment modalities. Int J Oral Maxillofac Surg 41:756-767.
59. Lau SL and Samman N (2006) Recurrence related to treatment modalities of unicystic ameloblastoma: a systematic review. Int J Oral Maxillofac Surg 35:681-690.
60. Al-Moraissi EA, Pogrel MA and Ellis E (2016) Enucleation with or without adjuvant therapy versus marsupialization with or without secondary enucleation in the treatment of keratocystic odontogenic tumors: A systematic review and meta-analysis. J Craniomaxillofac Surg 44:1395-1403.
61. Goodacre S (2015) Uncontrolled before-after studies: discouraged by Cochrane and the EMJ. Emerg Med J 32:507-508.
41
FIGURE LEGENDS
Figure 1 - Flow diagram of literature search and selection criteria (adapted from Preferred
Reporting Items for Systematic Reviews and Meta-Analysis).
TABLE LEGENDS
Table 1 - Descriptive characteristics of included articles; marsupialization technique (n=9).
Table 2 - Descriptive characteristics of included articles; decompression technique (n=18).
Table 3 - Risk of bias, assessed by the Joanna Briggs Institute Critical Appraisal Tools for
Figure 1 - Flow diagram of literature search and selection criteria (adapted from Preferred
43
Table 1 - Descriptive characteristics of included articles; marsupialization technique (n=9).
Author (Year); Country Sample (N) Number of cysts (n) Assessment method Intervention duration (mean±SD, range) Overall results
(mean±SD, range, or other pertinent findings) Kubota et al. (2013); Japan. N=72 (29F) n=72 cysts -DC=26 -OKC=28 -RC=18 Method: PR DC (11.0±5.0 mo) OKC (8.0±5.0 mo) RC (5.0±2.0 mo)
Mean relative speed of shrinkage (mm2/mo)
DC (7.0±6.0) OKC (7.0±3.0) RC** (3.4±0.7)
Time for radiolucent area shrank by half: DC (12.0±NR mo) OKC (9.0±NR mo) RC (6.0±NR mo) Miyawaki et al. (1999); Japan. N=20 (7F) n=20 cysts (DC) Method: PR
NR Speed of shrinkage (mm2/mo)
224.0±352.0 Nakamura et al. (2002a);
Japan. N=78 (32F) n=78 cysts (31 marsupialized) -MA=12 -UA=19 Method: PR MA=11.7±NR mo (3.0 to 22.0 mo) UA=21.3±NR mo (3.0 mo to 9.5 y) Nakamura’s rating Extremely effective (12.9%) Effective (61.3%) Ineffective (25.8%) Nakamura et al. (2002b); Japan. N=24 (10F) n=28 cysts (OKC) Method: PR 23.5±NR mo (6.0 to 80.0 mo) Reduction rate 81.4±NR (41.4 to 100%) Nakamura’s rating* Extremely effective (64.3%) Moderately effective (32.1%) Poorly effective (3.6%)
- 5 lesions did not need secondary surgery approach.
* Graded according to the author. Pogrel et al. (2004); United States of America. N=10 (4F) N=10 cysts (OKC) Method: PR
7.0 to 9.0 mo Initial maximal diameter (cm)
Range from 2.0 to 8.0
- All lesions completely resolved both clinically and radiographically. Shudou et al. (2012); Japan. N=15 (6F) n=15 cysts (OKC) Method: CT 413±NR d (136 to 1150 d)
Lesion volume OKC was reduce by: -50% in 239±NR d -75% in 478±NR d -87.5% in 717±NR d Yang et al. (2018); China. N=44 (19F) n=44 cysts -MA=33 -UA=11 Method: PR 16.6±NR mo (2.7 mo to 6.0 y) Reduction rate 65.6±NR% Effectiveness of marsupialization* - Extremely effective (59.1%) - Moderately effective (15.9%) - Poorly effective (9.1%) - Ineffective (15.9%)
*Graded according to the author Zhang et al. (2018); China. N=48 (27F) n=48 cysts (UA) Method: PR and CT 16.5±NR mo (6.0 to 20.8 mo) Reduction rate 36.9±NR% Nakamura’s rating Good (41.7%) Moderate (20.8%) Poor (37.5%) Zhao et al. (2011); China. N=53 (32F) n=53 cysts (OKC) Method: PR and injection volume assessment NR Reduction rate 79.7±NR% Cyst volume (ml) Preoperative (32.8±9.9) After 6 mo (6.7±2.8)
Legend: CT: computed tomography, d: days; DC: dentigerous cyst; F: female; MA: multicystic ameloblastoma; mo: months; NR: not reported; OKC: odontogenic keratocyst; PR: panoramic radiograph; RC: radicular cyst; SD: standard deviation; UA: unicystic ameloblastoma; y: years; **p<0.05.
Table 2 - Descriptive characteristics of included articles; decompression technique (n=18). Author (Year); Country Sample (N) Number of cysts (n) Assessment method Intervention duration (mean±SD, range) Overall results
(mean±SD, range, or other pertinent findings) Allon et al. (2015); Israel. N=26 (12F) n=32 cysts -DC=20 -OKC=9 -RC=3 Method: PR 7.4±2.6 mo (2.0 to 14.0 mo) Percentage of reduction 86.8±16.0% (49.0 to 100%) Nakamura’s rating Good (68.8%) Moderate (28.1%) Poor (3.1%)
- 17 lesions completely disappeared. Anavi et al. (2011); Israel. N=67 (28F) n=73 cysts -DC=28 -GOC=2 -OKC=22 -RC=17 -Unspecified=4 Method: PR 9.2±5.2 mo (2.0 to 33.0 mo) Percentage of reduction 79.3±NR% (28.0 to 100%) Nakamura’s rating Good (60.0%) Moderate (29.0%) Poor (11.0%) Asutay et al. (2016); Turkey. N=40 (17F) n=40 cysts -DC=12 -OKC=16 -UA=12 Method: CT
6.0±NR mo Mean percentage of volume decrease
48.2±12.2%
-In 28 lesions decompression was the definitive treatment. August et al. (2003); United States of America. N=14 (8F) n=14 (OKC) Method: PR 8.4±NR mo (6.0 to 12.0 mo)
Mean percentage of volume decrease 65.0±NR% (5.0 to 91.0%).
- 12 out of 14 cystic lesions presented more than 50% of shrinkage. Enislidis et al. (2004); Austria. N= 20 (14F) n= 20 cysts -DC=6 -OKC=8 -RC=5 Method: PR and CT 446±NR d (76 to 1379 d) Mean shrinkage 81.0±NR% Nakamura’s rating Good (n=13) Moderate (n=5) Poor (n=2) Gao et al. (2018); China. N=27 (11F) n=27 cysts -DC=15 -OKC=7 -UA=5 Method: CBCT 12.0±NR mo CBCT values (ΔΔCT) DC (138.9±50.3) OKC (165.7±67.0) UA (143.0±28.6) Gao et al. (2014); China. N=32 (11F) n= 32 cysts -OKC=20 -RC=4 -UA=8 Method: PR OKC (8.3±5.4 mo) RC (2.7±1 mo) UA (9.1±2.7 mo) Percentage of Reduction OKC (78.9±NR%) RC (79.6±NR%) UA (67.2±NR%)
Mean relative speed of shrinkage after decompression (cm2/mo) OKC (2.9±1.3) RC (3.4±0.7) UA (2.7±0.9) Jeong et al. (2017); South Korea. N=46 (13F) n= 46 cysts -DC=11 -NPC=4 -OKC=17 -POMC=2 -RC=7 -UA=5 Method: CT >6 mo (n=29)
≤6 mo (n=17) Nakamura’s rating Good (10.9%) Moderate (56.5%) Poor (32.6%) Reduction rate 57.9±NR% Lee et al. (2017); South Korea. N=17 (10F) n=17 cysts -DC=5 -OKC=10 -RC=1 -NPC=1 8.1±3.4 mo Nakamura’s rating Good (29.4%) Moderate (52.9%) Poor (17.7%) Reduction rate (%) 64±NR%
45 Method: PR -Diagnosis DC (62.6±NR%) OKC (70.0±NR%) RC (28.0±NR%) Lizio et al. (2018); Italy. N=25 (NR)
n=25 cysts (compatible with DC) Method: PR
20.0±NR mo Reduction rate
87.5±10.6% (63.4 to 98.8%)
- In 13 patients, the reduction was greater than 90%.
- No patient required a secondary surgery approach. Lizio et al. (2017); Italy. N=14 (3F) n=14 cysts (DC) Method: CBCT
8.0±NR mo Mean percentage of volume decrease
71.0±14.0% (54.0 to 95.0%) Lizio et al. (2013); Italy. N=20 (5F) n=20 cysts -DC=9 -OKC=10 -UA=1 Method: CBCT
5.7±NR (3.0 to 12.0 mo) Reduction rate
49.9±19.3% (0.4 to 98.7%)
- 18 patients underwent subsequent surgical enucleation of the lesion
Oh et al. (2018); South Korea. N=27 (11F) n=34 cysts (OKC) Method: PR and CT
9.8±NR (5.0 to 14.0 mo) Decreasing rate in maximum diameter 59.0±21.0%
Volume reduction rate 66.0±28.0% Oliveros-Lopez et al. (2017); Spain. N=23 (9F) n=23 cysts -DC=7 -RC=16 Method: PR
6.8±1.4 mo Monthly reduction rate (mm2/mo)
20.0±15.0 - Diagnosis DC (31.8±19.5) RC (13.8±6.5) Park et al. (2014); South Korea. N=32 (9F) n=32 cysts -DC=13 -OKC=14 -UA=5 Method: PR DC (6.9±NR mo) OKC (7.4±NR mo) UA (13.4 ±NR mo) Reduction rate - Diagnosis DC (39.4±NR%) OKC (33.6±NR%) UA (36.7±NR%) Park et al. (2019); South Korea. N=17 (9F) n=17 cyts -OKC=17 Method: CT
298±183 d Volume reduction rate
56.0±16.8% Song et al. (2015); South Korea. N=37 (11F) n= 37 cysts -DC=10 -OKC=17 -UA=10 Method: CT DC (194±81 d) OKC (250±180 d) UA (463±378 d)
Volume relative reduction - Diagnosis DC (48.5±24.5%) OKC (56.9±20.4%) UA (51.6±22.4%) Ulu et al. (2018); Turkey. N=13 (7F) N= 13 cysts -DC=1 -OKC=5 -RC=7 Method: CT 12.4±NR (6.0 to 18.0 mo) Initial volume (mm3) 7076.8±NR (3799.3 to 12471.3) - All patients recovered completely after decompression alone.
Legend: CT: computed tomography; CBCT: cone beam computed tomography; d: days; DC: dentigerous cyst; GOC: glandular odontogenic cyst; F: female; MA: multicystic ameloblastoma mo: months; NPC: nasopalatine duct cyst; NR: not reported; OKC: odontogenic keratocyst; POMC: postoperative maxillary cyst; PR: panoramic radiograph; RC: radicular cyst; SD: standard deviation; UA: unicystic ameloblastoma; y: years; **p<0.05.
