Presurgical Naso-Alveolar Molding (PSNAM) in Cleft Lip/Palate Neonates – A Bibliometric Analysis

Background: PSNAM is a paradigm shift from the conventional approach in the management of cleft lip/palate infants. However, bibliographic analysis of studies which are undertaken to examine research output or dissemination of research on PSNAM are lacking in the arena of Pediatric Dentistry.

Aim: To evaluate all the articles or studies published on the domain ‘presurgical naso-alveolar molding in cleft lip/palate infants.’

Design: A detailed search of bibliographic database was carried out for a period of two months from February to March 2023, using medical Mesh keywords. Each published articles were selected, tabulated and evaluated to obtain overall bibliographic details.

Results: A total of 69 articles were obtained following thorough search of the PUBMED database. Maximum publications were found in the journal Plastic Reconstructive Surgery Global Open. Ritschl LM contributed the greatest number of publications followed by Thakur S. India bagged with huge number of publications (26) followed by USA (14). Prospective studies were in maximum number followed by case-control studies.

Conclusion: There is a growing knowledge and application of PSNAM. However, there is insufficient scientific evidence containing randomized prospective studies pertaining to PSNAM. Future research and analysis of its application is highly warranted for its implementation in clinical practice.

Keywords: Bibliographic analysis, Cleft Lip/palate, Neonate, Presurgical Naso-alveolar molding, PSNAM

There is a paradigm shift in the treatment approach for the correction of cleft lip/palate in infants from traditional method to novel concept like presurgical naso-avleolar molding (PSNAM). This technique was introduced by Grayson which consists of active molding of alveolar process as well as the surrounding soft tissues and nasal cartilage. [1] This concept is a non-surgical method of reshaping the cleft lip, palate, alveolus and the nose to minimize the severity of the cleft deformity, before performing definitive surgical therapy in order to enhance the overall surgical outcome. It is associated with advantages like simplicity of the procedure and improving the quality of surgical repair, to obtain tension free muscle in cleft region. The primary goal of this technique in the patients with cleft lip/palate is to restore normal anatomy and function. [2]

Numerous articles including original studies, case reports and even systematic reviews have been published on the domain ‘use of PSNAM in cleft lip/palate infants’ in the arena of pediatric dentistry. [3-69] Although systematic reviews and randomized clinical trials (RCT) are rated as the most highly cited publications and represent research designs at the top of evidence hierarches, still they account only for 4-6% of research output. As a result, it is difficult for the implementation and dissemination of the research in order to examine ways of moving research evidence, guidelines and best practice recommendations into health practice.[70] A bibliographic review considers all the previous scientific knowledge of a given topic/domain about the research. It is a review article in itself, which can be published as such in scientific journals. Bibliographic reviews are frequently used to describe research activity and characterize research that is undertaken in terms of research output. Moreover, bibliographic studies and reviews have been carried out to describe public health research in broad sector. [71] Unfortunately, literature search did not reveal any such bibliographic reviews which have been undertaken to examine research output or dissemination of research in the field of PSNAM in cleft lip/palate infants. Therefore, the present bibliographic review was carried out to describe the research output pertaining to ‘application of PSNAM in cleft lip/palate infants.’

The current bibliographic analysis was conducted from the period September to October 2023 using available electronic data base which was retrieved and evaluated retrospectively. The present review used the guidelines of strengthening the Reporting of Observational Studies in Epidemiology (STROBE).

Inclusion criteria

The present bibliographic review included all type of studies, case reports and review articles and even surveys carried out on PSNAM in cleft lip and palate infants.

Exclusion criteria

Studies or case reports showing use of other preorthopedic appliances such as Hotz appliance, feeding appliances were excluded.

Study protocol

For a period of one month, a well-known electronic database like PubMed was searched using the medical Mesh keywords like pre-orthopedic treatment, cleft lip/palate, cleft infant, PSNAM, presurgical naso-alveolar molding, preorthopedic appliance with several combinations. All types of articles published in peer-reviewed journals irrespective of dental specialty were selected. Articles published only in English language were included irrespective of the date. Selected articles were read in detail focusing on title and abstract of the study. When additional information was required or in case of unclear picture of the study, the entire manuscript was downloaded and read. Published each article were tabulated covering the important details like article title, authors name, year of publication, journal name, country where study performed and type of the study.  Articles were tabulated in descending order based on the year of publication starting from new to old articles.

Following electronic search in well-known data base, PUBMED, a total of 451 articles were scrutinized. These articles were thoroughly searched, duplicates and irrelevant publications were eliminated based on the inclusion and exclusion criterion. Finally, only 69 articles were selected for the review and evaluation of which included all case reports, research studies and review articles. Articles with only abstracts including the above domain were not considered. Articles which found from the year 2005 to till date were arranged in descending order from the recent publication to older one (Table 1).

Table 1: List of selected articles in descending order from new to old showing title of the article, author’s name, year of publication, journal name and place of study done.

Descriptive analysis of publications

Publication year analysis

The first article about description of this new concept can be found in 2005 by Grayson and Maull. From 2005 onwards no publications were seen in the literature. Later again in 2009, Grayson and Shetye published a case report on this treatment modality. In 2010, from India, Mishra et al performed fist prospective clinical trial and published an article. From 2011 to 2023, numerous articles have been published in English language. Maximum publications can be seen in the year 2018 (n=10) followed by 2022 (8) and 2023 (n=7) (Table 2).

Table 2: Publication Year analysis

Author analysis

Analysis of authors who contributed to their publications in this domain consisted of 814 researchers. Published articles consisted authors in number ranging from a single author to maximum of 12 authors although there is a strict author guideline for the contribution to an original research study is about 6 authors and for a case report or review article is about 4 authors. This shows the quality of journals which incorporated more than six authors although they are known or listed for quality indexed journal in top most indexing database. Although maximum contribution of publications is from India (26), the credit of maximum number of publications is contributed by the author Ritschl LM (n=6) followed by Thakur S from India (n=5) and Broder HL (n=4). Authors like Thakur NS, Wolff KD, Singh A, Rani A, Diwana VK, Grill FD, Sischo L, Loeffelbein DJ and Grayson BH have contributed three publications each. Six articles were authored by only two authors and only four publications were authored by a single author (Table 3).

Table 3: Author’s analysis

Journal analysis

Among 69 articles, maximum publications were found in Plastic and Reconstructive Surgery Global Open journal (n=8), followed by Journal of Indian Prosthodontic Society (n=5). From other journals like Contemporary Clinical Dentistry, Indian Journal of Plastic Surgery and the Cleft Palate Craniofacial Journal, equal number of publications were retrieved (n=4). More than one articles were found in journals like Dental Research Journal (Isfahan), Clinical, Cosmetic and Investigational Dentistry, PLoS One, International Journal of Clinical Pediatric Dentistry, Journal of Craniofacial Surgery, Clinical Cosmetic Investigation Dentistry, Korean Journal of Orthodontics, Clinical Oral Investigation and Science Report journal (n=2) (Table 4).

Table 4: Journal analysis

Country analysis

Various nations across the globe have contributed publications about this domain. Among these, India is bagged with maximum publications (n=26) followed by USA (n=14), Germany (n=5) and Turkey (n=4). Other countries like Japan, Switzerland and China have contributed more than one publication (n=2). A single publication is given by countries like Iraq, Iran, Taiwan, Hungary, Peru, Brazil, Italy, Korea, SAU and Saudi Arabia (Table 5) (Figure 1).

Table 5: Country analysis

Figure 1: Pie-Diagram showing Publication contribution by different countries

Publication type analysis

Evaluation of publication type revealed 15 prospective studies, 10 case control studies, 7 retrospective studies, one randomized clinical trial, 15 case reports, 2 case series, 6 review articles, 7 surveys. 4 systematic reviews were found about this domain.  In addition to these, one letter to the editor and a single multi-centric study was found (Figure 2).

Figure 2: Analysis of different types of Publications on PSNAM

In this bibliographic review, all journals with keywords of PSNAM, NAM, cleft lip or palate, pre-orthopedic devices and which are published in English language in well-known database like PUBMED were searched from inception till august 2023.  All publications including original studies, review articles and even case reports were considered for evaluation. A total of 451 articles were found in database among which only 69 articles were selected based on the inclusion criteria. The selected 69 articles were classified as original studies, review articles and case reports and systematic reviews. In terms of period of publication, articles can be seen from 2005 to 2023. Although this concept has been suggested long back in the year 2005, till 2011, except one case report and a review article, no original studies publications can be found in the literature. Maximum publications were found in the year 2018 (n=10). In the year 2019 a tremendous decline in publication/research (n=2) can be found which may be due to COVID attack. When type of publications was analysed, it was found that 15 were case reports and two were case series, six were review articles, four of systematic review seven were of questionnaire surveys and remaining were original studies including prospective, case-control, RCT and retrospective studies. Though these original studies showed high level of evidence and impact of the article, however its relevance to further research is highly warranted.  One landmark publication was noticed in this analysis which includes a retrospective multi-center study evaluating the intercultural parental stress caused by PSNAM therapy conducted by Roth and his co-researchers in 2021.

Analysis of publication contribution by different countries towards PSNAM revealed majority of articles by institutions and authors from India (n=26). The reason behind this fact can be attributed to the increased prevalence of consanguineous marriages and in turn occurrence of high number of cleft lip and palate births, vast population, focus on health sciences and research, and promotion of health research through the availability of free treatment organizations like ‘Smile train’ as well as government funding. In addition to these, recently India is tremendously improving by leaps and bounds in terms of development and medical research. The second highest number of publications were contributed from USA (n=14) followed by Germany (n=5) and Turkey (n=4). Japan, China and Switzerland contributed two publications each pertaining to PSNAM.

Author’s contribution towards PSNAM publications showed maximum publication of six by Ritschl from Germany. However, among these six publications, in only one publication he is authored as first author. In contrast to this, the second maximum publications were evidenced from India by author Thakur S bagged with 5 publications. Among these five publications, he is listed as first author in four publications. Author Broder HL from Germany contributed four publications. Articles with single author are contributed by four people like Jain, Datta, and Rajguru all from India37, and Altug42 from Turkey. Publications authored by only two authors were found in six articles. They are Gonca and Ozel from Turkey, Denadai and Lo from Brazil, Jayashree and Paul from India, Grayson wit Shetye from New York, Esenlik and Aydin from Turkey and Grayson and Maull from New York. Six publications were authored by three authors. Great number of publications (n=8) were found in the journal Plastic and Reconstructive Surgery Global Open which has impact factor of 3.6 followed by Journal of Indian Prosthodontic Society (n=5) with an impact factor 1.2. Journals like Contemporary Clinical Dentistry, The Cleft Palate-Craniofacial Journal and Indian Journal of Plastic Surgery have contributed four publications each.

From the current bibliographic analysis regarding application of PSNAM in cleft lip/palate infants, few limitations were observed. Although author have used thorough and systematic method for identification, selection and analysis of all articles published so far on PSNAM in cleft lip/palate domain, a single data base search (only PUBMED) was used in this evaluation. Therefore, articles or publications on the same domain with different titles or with different combination of keywords will be obtained from search of other databases like Scopus, Web of Science, Google Scholar or Embase.  Another limitation experienced was author details were missing in some publications and it was difficult to trace the primary author who contributed to the particular study in case of multiple authors. Therefore, author with corresponding address or corresponding author was considered as primary author. The present review definitely marks an important source of information for other researchers to conduct such more reviews.

Finally, authors would like to conclude that the present bibliographic analysis on the domain ‘application of PSNAM in cleft lip/palate infants’ identified numerous different publications. There is a growing knowledge and application of PSNAM in cleft lip/palate infants. However, there is insufficient scientific evidence containing randomized prospective studies pertaining to PSNAM. Future research and analysis of its application is highly warranted for its implementation in clinical practice.

Why this paper is important to paediatric dentists

• Pediatric dentist should focus on this treatment modality by implementing this novel approach in the treatment of cleft lip/palate infants.
• The interdepartmental collaboration including multiple specialities should happen to enhance research output.
• The present analysis provides evidence and research output across the globe on existing literature of PSNAM in cleft lip/palate infants.