Publication Trends in Craniofacial Cleft Reconstruction: A PubMed-Based Bibliometric Analysis of the Literature from 2000 to 2025

Introduction

Cleft lip and palate are among the most common congenital anomalies affecting the orofacial region and require coordinated multidisciplinary care across surgical and allied specialties. ¹ Craniofacial clefts represent a rarer and more complex group of deformities, and their classification and anatomic interpretation have been strongly shaped by Tessier-based concepts and neuroembryologic understanding. ²

Bibliometric analysis is a useful method for evaluating the structure and evolution of a scientific field. Recent methodological papers have described practical workflows for performing bibliometric studies using database-derived records and dedicated software platforms such as Biblioshiny, Bibliometrix, and VOSviewer.^3,4

Within cleft and related maxillofacial literature, bibliometric studies have already been used to examine influential publications, regional publication trends, and research development over time.^5-7 These studies show that bibliometric methods can identify patterns in authorship, institutional productivity, citation impact, and thematic focus.

At the same time, reconstructive care in cleft and craniofacial surgery spans multiple domains, including alveolar cleft grafting, multidisciplinary management, and Tessier craniofacial cleft reconstruction.^8-10 Despite this breadth, the literature specifically related to craniofacial cleft reconstruction has not been synthesized bibliometrically in a focused manner.

The present study was therefore designed to perform a bibliometric analysis of the literature on craniofacial cleft reconstruction published from 2000 to 2025. The objectives were to evaluate publication trends over time and describe the distribution of journals, study types, reconstructive themes, anatomic focus, and reported follow-up patterns within this field.^3,4

Methods

Data Extraction and Bibliometric Analysis

Bibliographic information was extracted from the included PubMed records for descriptive bibliometric evaluation. The analysis examined publication output and the distribution of journals, study types, reconstructive themes, and follow-up patterns within the dataset. Methodological guidance for bibliometric analysis and the use of Biblioshiny/Bibliometrix and VOSviewer was drawn from published bibliometric workflow papers.^3,4 Extracted clinical and methodological characteristics were tabulated descriptively, including study design, level of evidence, cleft/fissure type, anatomic focus, treatment approach, staging, follow-up reporting, and outcome domains (Figures 1 and 2).

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Figure 1.

Study selection flow diagram. Flow diagram showing identification and screening of PubMed records for inclusion in the bibliometric dataset. Fifty-seven records were screened by title and abstract, 16 were excluded, and 41 articles were included in the final analysis.

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Figure 2.

Year-wise publication trend. Annual distribution of included publications on craniofacial cleft reconstruction from 2003 to 2025.

Results

The PubMed search yielded 57 records after application of the predefined search strategy and filters. Title and abstract screening resulted in 41 articles being included in the final bibliometric dataset.

Eligible publications were distributed unevenly across the study period. The earliest included article was published in 2003, and the highest annual output was observed in 2014 with 5 publications. Smaller peaks were seen in 2005, 2008, 2019, and 2023 with 3 publications each.

The included literature was dominated by descriptive clinical reports, particularly case reports, case series, and single-center surgical experiences, whereas larger cohort analyses were uncommon.

The publications were dispersed across several specialty journals, with a clear concentration in craniofacial, plastic surgery, and cleft-focused journals. Journal of Craniofacial Surgery was the most frequent publication venue with 14 articles, followed by Cleft Palate-Craniofacial Journal with 7 and Journal of Cranio-Maxillofacial Surgery with 6. Ophthalmic Plastic and Reconstructive Surgery and Annals of Plastic Surgery each contributed 3 articles (Tables 1 and 2).

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Table 1.

Year-Wise Distribution of Included Publications on Craniofacial Cleft Reconstruction (2000-2025).

Year	Number of Publications
2003	1
2004	0
2005	3
2006	0
2007	1
2008	3
2009	1
2010	2
2011	0
2012	2
2013	2
2014	5
2015	2
2016	1
2017	2
2018	2
2019	3
2020	0
2021	4
2022	1
2023	3
2024	1
2025	2
Total	41

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Table 2.

Journal-Wise Distribution of Included Publications.

Journal	Number of Publications
Journal of Craniofacial Surgery	14
Cleft Palate-Craniofacial Journal	7
Journal of Cranio-Maxillofacial Surgery	6
Ophthalmic Plastic and Reconstructive Surgery	3
Annals of Plastic Surgery	3
Journal of Plastic, Reconstructive & Aesthetic Surgery	2
Plastic and Reconstructive Surgery	2
Craniomaxillofacial Trauma and Reconstruction	1
Annals of Maxillofacial Surgery	1
Polish Journal of Surgery (Pol Przegl Chir)	1
Facial Plastic Surgery Clinics of North America	1
Total	41

Anatomically, the included studies addressed a broad range of craniofacial cleft presentations, with repeated attention to Tessier no. 3 and no. 4 clefts, midline clefts, nasal clefts, orbital and eyelid involvement, and lateral facial clefts or macrostomia. Reconstructive themes included local and regional flap-based repair, facial unit repositioning, nasal and alar reconstruction, eyebrow and eyelid reconstruction, bone grafting, distraction-assisted treatment, and staged protocol-based correction. The extracted clinical and methodological characteristics of the included publications are summarized in Table 3.

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Table 3.

Extracted Clinical and Methodological Characteristics of Included Craniofacial Cleft Reconstruction Publications.

Extracted Domain	Categories Identified in the Included Literature	Summary Pattern Observed
Study design/article type	Case reports; small case series; retrospective institutional experiences; protocol-based surgical reports; technical or management-focused reviews	The literature was dominated by descriptive clinical reporting. Larger comparative studies, multicenter datasets, and high-level analytic designs were uncommon.
Level of evidence	Predominantly low-level clinical evidence; mainly individual cases, small series, and expert/institutional experience	The evidence base reflected the rarity and heterogeneity of craniofacial clefts. Most publications contributed surgical experience rather than comparative effectiveness data.
Cleft/fissure type	Tessier no. 0; Tessier no. 0–14; Tessier no. 3; Tessier no. 4; Tessier no. 5; Tessier no. 7; Tessier no. 10; median and paramedian craniofacial clefts; lateral facial clefts/macrostomia; nasal clefts; orbital and eyelid clefts	Repeated attention was seen for midline, orbital, nasal, and lateral facial cleft patterns. Tessier no. 3, no. 4, no. 7, and midline 0–14 clefts appeared as recurring reconstructive themes.
Main anatomic region involved	Orbit; eyelids; eyebrow; nose; alar region; upper lip; maxilla; midface; frontal bone; cranial vault; lateral oral commissure	The included literature involved both soft-tissue and skeletal deformities, often spanning multiple facial units in the same patient.
Treatment/reconstruction approach	Local and regional flap repair; nasal and alar reconstruction; eyelid and eyebrow reconstruction; facial unit repositioning; bone grafting; craniofacial osteotomy; hypertelorism correction; distraction-assisted treatment; staged reconstruction; multidisciplinary protocol-based management	Reconstruction was usually individualized according to cleft anatomy. Many reports emphasized staged correction rather than a single definitive operation.
Timing/staging of care	Primary repair in infancy or childhood; staged childhood correction; post-growth correction; adult secondary reconstruction; long-term revision surgery	Several reports described growth-dependent sequencing of reconstruction, especially for midline, orbital, and major craniofacial clefts.
Follow-up reporting	Immediate postoperative outcome; short-term follow-up; long-term follow-up into adolescence or adulthood; protocol completion from birth to maturity	Follow-up reporting was inconsistent. Some papers reported only immediate or short-term outcomes, while others described prolonged staged care and late stability.
Outcome domains reported	Aesthetic correction; facial symmetry; orbital position; eyelid function; nasal form; oral commissure position; skeletal stability; relapse; need for secondary revision; functional improvement where applicable	Outcome reporting was mostly descriptive. Standardized quantitative outcome measures were rarely emphasized across the literature.
Specialty/team involvement	Plastic surgery; craniofacial surgery; maxillofacial surgery; ophthalmic plastic surgery; otolaryngology; neurosurgery; multidisciplinary craniofacial teams	Complex clefts frequently required multispecialty input, particularly when orbital, nasal, cranial, or hypertelorism correction was involved.
Main limitation visible across included studies	Small sample size; heterogeneity of cleft anatomy; variable follow-up; non-standardized outcome reporting; limited comparative data	The overall evidence base remained fragmented and difficult to compare across studies, supporting the need for standardized reporting and collaborative datasets.

Follow-up reporting was variable across the included studies. Some publications described only immediate reconstructive outcomes, whereas others reported staged management across childhood or long-term follow-up extending into adolescence or adulthood.

Overall, the bibliometric dataset demonstrated that the published literature on craniofacial cleft reconstruction from 2000 to 2025 was limited in volume, heterogeneous in anatomic focus, and dominated by descriptive surgical reporting.

Discussion

The present bibliometric analysis identified a relatively small but persistent body of literature on craniofacial cleft reconstruction between 2000 and 2025. The included publications were predominantly descriptive surgical reports, and comparative or higher-level analytic studies were uncommon. This pattern is consistent with the rarity and heterogeneity of craniofacial clefts and also aligns with prior bibliometric observations in cleft-focused and related craniomaxillofacial literature, where research output has been shaped largely by specialized centers, experience-based reporting, and uneven publication volume.^5-7

The predominance of descriptive studies in the present dataset is also consistent with the broader evidence structure of cleft and craniofacial surgery. A review of randomized controlled trials has shown that Level I evidence in this field remains limited, with many commonly performed procedures not supported by robust comparative trial data. ¹¹ In rare facial and craniofacial clefts specifically, published experience has often come from institutional series and protocol-based clinical reports rather than large multicenter studies. ¹²

The clinical breadth of craniofacial cleft reconstruction also helps explain why the literature remains heterogeneous. Reconstructive problems in this field range from alveolar cleft grafting and broader cleft-craniofacial surgical management to the varied anatomic patterns described within Tessier clefts, making uniform comparison across studies inherently difficult.^8-10

Rare craniofacial clefts have been described as an underreported group of deformities, and objective long-term evaluation has often been difficult because of their rarity and phenotypic diversity. ¹³ Bonafos et al. also noted that the published literature in rare craniofacial clefts is sparse and frequently limited to isolated cases or very small series. ¹⁴ In a retrospective series of 32 Tessier no. 0 clefts, da Silva Freitas et al. likewise noted a paucity of publications on this malformation and observed that the available literature was largely confined to clinical examination and surgical treatment in small numbers of patients. ¹⁵ In an 11-patient series, Fijałkowska and Antoszewski described treatment as multispecialist, multistage, and highly individualized. ¹⁶ Monasterio and Taylor similarly argued that standard techniques are difficult to propose for major craniofacial clefts because of the many possible combinations of skeletal and soft-tissue involvement. ¹⁷

This need for individualized planning is evident in papers that propose cleft-specific treatment protocols. Woods et al. described an age-based management protocol for Tessier no. 7 clefts, separating infancy, childhood, and post-growth interventions. ¹⁸ Golinko et al. reported a four-step primary surgical approach for Tessier no. 3 clefts and anticipated later lip, nose, eyelid, maxillary, orbital, and alveolar revisions. ¹⁹ Balaji also emphasized that rare facial Tessier clefts often require more than one operation and do not lend themselves easily to a single universal treatment algorithm. ²⁰

Long-term follow-up is especially important because reconstructive adequacy cannot be judged from the immediate postoperative appearance alone. Toriyama et al. reported stable long-term reconstruction over 7 years in a Tessier no. 10 craniofacial cleft treated with folded vascularized calvarial bone. ²¹ Afifi et al. described more than a decade of additional reconstruction in an adult with a Tessier no. 5 facial cleft who had already undergone numerous prior procedures. ²² In the protocol series of midline Tessier 0–14 clefts, Pidgeon et al. followed patients from birth to maturity and highlighted the importance of growth-dependent sequencing of correction. ¹³

Periorbital and midline deformities add another layer of complexity to craniofacial cleft reconstruction. Rüegg et al. managed median and paramedian craniofacial clefts using procedures such as box osteotomy, facial bipartition, canthopexy, and nasal reconstruction according to the underlying deformity pattern. ²³ Pittet et al. reported that simultaneous correction of Tessier 0–14 deformity with major hypertelorism, frontal bone defect, and nasal aplasia required repeated operations and neurosurgical participation. ²⁴ Golinko et al. likewise used a coordinated approach involving plastic surgery, otolaryngology, and oculoplastic surgery for Tessier no. 3 repair. ¹⁹

Even when skeletal correction is achieved, durability over time remains a concern. In a long-term follow-up study of hypertelorbitism correction, Wan et al. found greater relapse when correction was performed at a younger age. ²⁵ This point is relevant when interpreting reconstructive stability in craniofacial cleft patients who undergo orbital and midfacial correction during growth.

Against this clinical background, the pattern seen in the present bibliometric dataset becomes easier to understand. A field characterized by rarity, marked anatomic variability, multistage treatment, and prolonged follow-up is more likely to generate case reports, case series, protocol papers, and single-center experiences than large comparative trials. The extracted-characteristics summary further demonstrates that the literature is not only numerically limited but also methodologically heterogeneous, with variable reporting of cleft type, reconstructive approach, staging, follow-up, and outcome domains.

Several limitations of the present study should be acknowledged. The dataset was derived from a single database and was restricted to English-language human studies. Study selection was based on title and abstract screening rather than full-text review of every retrieved record. In addition, the search strategy was intentionally focused on Tessier and craniofacial cleft terminology, so relevant studies using different nomenclature may not have been captured.

Despite these limitations, the present analysis provides a focused overview of how the craniofacial cleft reconstruction literature has developed between 2000 and 2025. The available evidence remains limited in volume and is dominated by descriptive surgical reporting, but it also shows sustained effort by specialized centers to refine reconstructive strategies for these rare deformities. Future work would benefit from standardized outcome reporting, longer structured follow-up, and greater collaboration across centers treating craniofacial clefts.