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Systematic Review

Comparison of Surgical Techniques in Managing Craniosynostosis: Systematic Review and Bayesian Network Meta-Analysis

by
Muhammad Ikhlas Abdian Putra
1,
Mirnasari Amirsyah
1,*,
Budiman Budiman
2,
Shakira Amirah
3,
Seba Talat Al-Gunaid
4 and
Muhammad Iqhrammullah
5,*
1
Plastic Reconstructive and Aesthetic Surgery Subdivision, Department of Surgery, Zainoel Abidin General Hospital, Medical Faculty of Universitas Syiah Kuala, Banda Aceh 23111, Indonesia
2
Plastic Reconstructive and Aesthetic Surgery Subdivision, Department of Surgery, Gatot Soebroto Army Central Hospital, Medical Faculty of Universitas Syiah Kuala, Jakarta 10410, Indonesia
3
Faculty of Medicine, Universitas Indonesia, Jakarta 10430, Indonesia
4
School of Medicine, Universitas Syiah Kuala, Banda Aceh 23243, Indonesia
5
Postgraduate Program of Public Health, Universitas Muhammadiyah Aceh, Banda Aceh 23123, Indonesia
*
Authors to whom correspondence should be addressed.
Surgeries 2024, 5(4), 970-985; https://doi.org/10.3390/surgeries5040078
Submission received: 26 September 2024 / Revised: 9 October 2024 / Accepted: 23 October 2024 / Published: 28 October 2024
Browse Figures
Versions Notes

Abstract

:
Open total cranial vault reconstruction (CVR) is the common procedure in managing craniosynostosis, yet more techniques have been introduced as alternatives, namely endoscopic suturectomy (ES), endoscopy-assisted craniectomy (EC), spring-assisted surgery (SAS), strip craniectomy with helmet (SC), Pi craniectomy (PiC), Pi plasty (PiP), and Renier’s “H” technique (RH). The aim of this study was to compare the effectiveness of craniosynostosis surgeries in improving the cephalic index of the patients. Studies published until 7 March 2024 reporting CVR, ES, SAS, SC, RH, and PiP as definitive craniosynostosis management with the cephalic index as the outcome were included. Bayesian network meta-analysis and pair-wise meta-analysis were performed using a random-effects model based on standardized mean difference (SMD) and 95% confidence interval (CI). Nine studies published in 2008–2024 recruiting a total of 464 craniosynostosis patients (age: 18–61 months) were included in this meta-analysis. EC (SMD = 0.23 [95%CI: −5.47 to 5.63]; p = 0.935), PiP (SMD = −0.07 [95%CI: −9.27 to 8.79]; p = 0.988), ES (SMD = −0.59 [95%CI: −6.07 to 4.94]; p = 0.834), PiC (SMD = −1.16 [95%CI: −8.89 to 6.35]; p = 0.765), RH (SMD = −0.96 [95%CI: −6.62 to 4.53]; p = 0.736), SAS (SMD = −0.86 [95%CI: −8.25 to 6.18]; p = 0.815), and SC (SMD = −1.79 [95%CI: −9.05 to 5.28]; p = 0.624) were found to be as effective as CVR in improving the cephalic index. Network meta-analysis suggests that PiP is the most effective among these techniques (rank 1 probability = 0.273). According to the rank probabilities of our model the order of techniques from the most to the least effective is as follows: EC > CVR > PiP > ES > SAS > RH > PiC > SC.

1. Introduction

Craniosynostosis is a condition characterized by the premature fusion of one or more cranial sutures, which restricts skull growth and may result in abnormal cranial morphology [1]. This condition shares similarities with other causes of skull malformation, such as positional plagiocephaly and other forms of sutural synostosis [2]. These conditions are influenced by factors like asymmetric growth and sutural displacement, while additional contributors such as acquired diseases and skeletal dysplasias, including osteopetrosis and frontometaphyseal dysplasia, can further alter skull structure and function [2]. The impacts of the skull malformation may include increased intracranial pressure, abnormal head shape, and potential neurodevelopmental delays [1]. Indeed, a meta-analysis suggested the absence of statistical significance in the correlation between craniosynostosis and attention deficit/hyperactivity disorder [3]. However, the limited studies gathered in that systematic review suggest the evidence remains, in fact, inconclusive [3,4]. The global prevalence of craniosynostosis is estimated to be about 5.9 cases per 10,000 live births [5]. However, there are regional differences in prevalence. In Africa, for instance, the estimated incidence is 4 cases per 10,000 live births [6].
Craniosynostosis can be classified as syndromic or non-syndromic [1]. Syndromic craniosynostosis presents with other congenital malformations or developmental delays, while non-syndromic cases occur in isolation without other anomalies [7]. Over 75% of craniosynostosis cases are non-syndromic [8]. Syndromic craniosynostosis is often linked to genetic mutations, such as those seen in Apert, Crouzon, and Pfeiffer syndromes [9]. Different cranial shape types are associated with these syndromes, with hyperbrachycephalic shapes being more frequent in Apert syndrome, while mesocephalic shapes are more typical in Crouzon syndrome [10,11]. Non-syndromic cases are usually sporadic, with a multifactorial origin that includes genetic and environmental factors [12,13]. Approximately 85% of craniosynostosis cases are non-syndromic, with several genetic variants, particularly in genes like SMAD6, contributing to an increased risk [12]. Environmental factors such as intrauterine head constraint, oligohydramnios, and exposure to teratogens may also play a role in its development [13,14].
Surgery is the main treatment for craniosynostosis. Open cranial vault remodeling (CVR) is one of the most common and well-known techniques [15]. CVR involves removing, reshaping, and repositioning the cranial bones to correct the skull deformities, followed by securing the repositioned segments with internal fixation devices such as absorbable plates and screws [15]. This approach is generally recommended for patients older than six months or when less invasive techniques, like endoscopic-assisted craniectomy (EC), are unsuitable due to the severity of the deformity or the child’s age [16]. CVR is favored for its ability to achieve a significant degree of correction; however, it is more invasive and is associated with a higher risk of complications, such as increased operative time (approximately 4 to 6 h) and blood loss (averaging 200–500 mL), compared to minimally invasive techniques [17]. Early surgical intervention, ideally within the first year of life, leads to better outcomes due to the brain’s rapid growth phase and the skull’s ability to remodel itself [16]. Late treatment is often associated with more complex surgeries, longer hospital stays, higher costs, and increased risks of complications [16]. Additionally, in those receiving an early procedure, it has a significantly higher impact on quality of life, influencing psychological well-being, social functioning, and physical health [18].
Several alternative techniques have been documented for the treatment of craniosynostosis, including endoscopic suturectomy (ES), endoscopy-assisted craniectomy (EC), spring-assisted surgery (SAS), strip craniectomy with helmet (SC), Pi craniectomy (PiC), Pi plasty (PiP), and Renier’s “H” technique (RH) [19]. While some studies have reported that minimally invasive endoscopic techniques such as ES and EC result in reduced blood loss, shorter surgical time, and lower transfusion rates compared to traditional open methods [20,21,22], the comparative effectiveness of these surgical approaches remains undetermined due to substantial variability in outcomes across different studies [23]. Prior meta-analyses on craniosynostosis surgeries have utilized a pair-wise model [24,25], which limits comparisons to certain techniques only. To close this knowledge gap, this study performed a Bayesian network meta-analysis to compare the reported outcomes of different craniosynostosis surgeries comprehensively.
One of the most important outcomes in craniosynostosis surgeries is the cephalic index, which is an anthropometric measurement used to monitor changes in the shape of the head. The cephalic index is an anthropometric measurement calculated by multiplying the maximum width of the head by 100 and dividing it by the maximum length of the head. It serves solely as a continuous variable to estimate the size effect in this meta-analysis, without applying any further classification categories. The cephalic index was chosen as the main outcome in the present study because it is considered a standardized assessment in craniosynostosis surgeries, could be used for the prognosis of the surgery, and allows for long-term follow-up (as compared to other parameters such as hospital stay or blood transfusion rate, which can only act as short-term parameters) [26,27]. Moreover, the parameter is generally independent of the presence or absence of symptoms related to craniosynostosis allowing the comparability between groups.

2. Method

2.1. Study Design

Prior to performing this review, a set of protocols was designed in accordance with the PRISNMA (Preferred Reporting Items for Systematic Reviews and Network Meta-Analyses) guidelines [28]. The research question was “are alternatives for CVR more effective in improving cephalic index among patients with craniosynostosis”? Protocol registration was carried out retrospectively on Open Science Framework Registries, and the protocol can be accessed at https://doi.org/10.17605/OSF.IO/EHXVM (accessed on 11 October 2024).

2.2. Search Strategies

A literature search was performed on 7 March 2024 by utilizing search engines in the following databases: PubMed, Scilit, Scopus, and Web of Science (WoS). The Boolean operators “AND” and “OR” were used. Complete details of the keyword combinations used in the different databases are presented in Table 1.

2.3. Inclusion and Exclusion Criteria

Studies reporting the definitive surgeries for craniosynostosis management (i.e., CVR, ES, SAS, SC, RH, and PiP) and the cephalic index as the outcome were included. The inclusion criteria covered all types of craniosynostosis (i.e., sagittal, metopic, coronal, and multiple sutures), either syndromic or non-syndromic. Since the network meta-analysis does not require a control, we did not determine which technique acted as the control in the eligibility criteria. Eligible study designs were cross-sectional, case–control, and cohort. Case–control or case series were excluded. Review articles, commentaries, editorials, and conference abstracts were excluded.

2.4. Screening and Selection

Following the automatic removal of duplicates in EndNote 19, the screening was performed in two stages: the first stage was based on abstract and title, whilst the second stage was based on full texts. Both stages were carried out by two independent review authors (S.A. and S.T.A.G.). Discrepancies were resolved through consensus; if consensus was not reached, a consultation with the third review author (M.I.) was performed.

2.5. Critical Appraisal

Two independent review authors (M.I. and M.I.A.P.) independently assessed the quality of the included studies. The Newcastle–Ottawa Scale (NOS) for observational studies was used for the critical appraisal. A description of this tool has previously been reported in detail [29]. Any discrepancies were resolved through consensus or consultation with the third review author (S.A.).

2.6. Data Extraction

Firstly, the characteristics of the included studies, namely the first author’s name, year of publication, study design, and sample size, were extracted. The demographic data of the research subjects were then extracted and included age (months) and gender (male/female). Clinical characteristics, such as affected sutures (sagittal, coronal, metopic, lambdoid, or mixed) and “syndromic or not” status, were also collected. The post-surgical cephalic index was extracted as the outcome. Continuous data were presented as mean ± standard deviation (SD); otherwise, conversion was made in using the suggested methods from previous studies [30,31,32]. Data extraction was performed by M.I.A.P. and reconfirmed by the second review author (M.I.).

2.7. Statistical Analysis

A pair-wise meta-analysis was carried out on RevMan 5.4 for surgical techniques with the outcome (cephalic index) reported more than once. Network meta-analysis based on Bayesian statistics was performed for all surgical techniques using the gemtc package in Rstudio version 2024.04.2 (Posit PBC, Boston, MA, USA). A random-effects model was chosen for both the pair-wise and network meta-analyses, where the pooled size effect was calculated using the standard mean difference (SMD). Heterogeneity in pooled estimates was observed based on I2 > 50% or p-Het < 0.1. Ninety-five percent confidence intervals (95%CIs) were also calculated. Publication bias was observed through funnel plots only if the number of publications was over 10.

3. Results

3.1. Search Results

The initial screening resulted in 1473 records identified across the four databases, whereas duplication removal left 569 for abstract and title screening. As many as 102 records were sought for full-text screening, but only 99 of these had accessible full texts. We then found that 6 records were duplicates, 24 records had no comparison, 9 records were review articles, 45 records reported outcomes other than the cephalic index, and 6 records were case reports or case series; hence, these were excluded. Finally, we found nine studies eligible for qualitative and quantitative review [20,21,33,34,35,36,37,38,39]. The PRISMA flow diagram depicting the overall process of the screening and selection is presented in Figure 1.

3.2. Characteristics of Included Studies

The characteristics of the included studies are presented in Table 2. All studies had a higher number of male patients than female patients, with patients’ ages ranging from 2.9 ± 2.8 to 13.4 ± 19.2 months [20,21,33,34,35,36,37,38,39]. Almost all studies reported sagittal craniosynostosis [21,33,34,35,36,37,38], though one study reported mixed sutures and another did not report the affected suture [20]. Six studies reported the outcome exclusively in non-syndromic patients [21,33,34,35,37,39].

3.3. Quality of Included Studies

Almost all studies presented adequate case definitions and descriptions of the used surgical techniques [34,35,36]. As many as three studies did not describe the surgical techniques used [21,33,38]. Six studies had an imbalanced number of patients between the CVR and non-CVR groups [20,21,33,34,35,36,38]. Additionally, one study also did not exclusively include non-syndromic patients, which could have contributed to heterogeneity [21]. Due to the low number of patients, the analysis of the outcome of one study is considered to be non-robust [36]. The summary of the critical appraisal results for the included studies is presented in Table 3.

3.4. Network Meta-Analysis

We found six surgical techniques used to manage craniosynostosis. The network model established to compare each technique is presented in Figure 2. Direct connections were established for CVR versus EC, CVR versus ES, CVR versus RH, SAS versus PiP, SC versus EC, EC versus PiC, SC versus SAS, SAS versus EC, and SAS versus PiP. Other comparisons were estimated through indirect connections. The results of the comparison based on the network meta-analysis are presented in Table 4.
A forest plot depicting the total effect of each technique when CVR is chosen as the comparator is presented in Figure 3. The highest SMD was found in EC (SMD = 0.23 [95%CI: −5.47 to 5.63]; p = 0.935), followed by PiP (SMD = −0.07 [95%CI: −9.27 to 8.79]; p = 0.988). Other techniques, such as ES, PiC, RH, SAS, and SC, had SMDs of −0.59 (95%CI: −6.07 to 4.94; p = 0.834), −1.16 (95%CI: −8.89 to 6.35; p = 0.765), −0.96 (95%CI: −6.62 to 4.53; p = 0.736), −0.86 (95%CI: −8.25 to 6.18; p = 0.815), and −1.79 (95%CI: −9.05 to 5.28; p = 0.624), respectively.

3.5. Rank Probabilities

The rank probabilities of craniosynostosis surgeries improving the cephalic index, as estimated through the Bayesian approach, are presented in Table 5. PiP has the highest score (0.273) in the first rank, while EC has the highest score (0.246) in the second rank. As for the third and fourth ranks, the highest scores are exhibited by CVR (0.201) and SAS (0.155), respectively. According to the weighted scores, the following order was found: EC (3.01) > CVR (3.48) > PiP (3.72) > ES (4.48) > SAS (4.92) > RH (5.02) > PiC (5.23) > SC (6.16).

3.6. Publication Bias

Publication bias was not assessed because the number of included studies did not reach the minimum threshold (n = 10) [40].

4. Discussion

4.1. Surgical Techniques for Craniosynostosis Management

A systematic search of the predetermined strategies resulted in studies reporting the use of seven different surgical techniques for craniosynostosis management. These techniques include open total cranial vault reconstruction (CVR), endoscopic suturectomy (ES), endoscopy-assisted craniectomy (EC), spring-assisted surgery (SAS), strip craniectomy with helmet (SC), Pi craniectomy (PiC), Pi plasty (PiP), and Renier’s “H” technique (RH). A summary of the key features, indications, and reported outcomes of each surgical technique is presented in Table 6.

4.1.1. Open Total Cranial Vault Reconstruction

CVR is a surgical procedure for patients with complex or multi-suture craniosynostosis, particularly when less invasive methods do not provide sufficient correction [36,41]. The procedure involves the removal, reshaping, and repositioning of cranial bones to increase intracranial volume and achieve a normal skull shape. The techniques used in CVR include creating large cranial flaps that are reshaped to accommodate brain growth and secured with bioabsorbable plates and screws to maintain the new configuration [48,49]. This method effectively corrects abnormal cranial shapes and relieves elevated intracranial pressure, offering both functional and aesthetic benefits. CVR is often associated with a longer recovery period and carries risks such as blood loss and infection [50,51]. Studies show that CVR prevents neurocognitive issues by allowing proper brain expansion and improves psychological and social outcomes through the normalization of head shape [52]. Due to its ability to address both structural and developmental concerns, CVR remains the preferred approach for managing severe craniosynostosis [20].

4.1.2. Endoscopic Suturectomy

ES is a minimally invasive surgical technique used to treat craniosynostosis, particularly suitable for infants with single-suture synostosis [23]. The procedure involves making small incisions through which an endoscope is inserted to remove the fused suture. This approach minimizes blood loss and reduces the length of hospital stay compared to traditional open surgeries [20,42,45]. Following the surgery, helmet therapy is typically recommended to guide the skull’s reshaping and to encourage symmetrical cranial growth as the brain continues to expand [34]. The advantages of an endoscopic suturectomy include less scarring, faster recovery, and a reduced risk of complications, although it is less applicable in cases involving multiple sutures or older children [20]. Common outcomes include an effective reshaping of the skull and improved cranial symmetry, with lower risks of infection and shorter healing times than with open surgery [51].

4.1.3. Endoscopy-Assisted Craniectomy

EC is a minimally invasive surgical technique designed to treat craniosynostosis by removing the sections of the skull where premature suture fusion has occurred. The primary goal of this technique is to release the fused suture, enabling the skull to expand and accommodate normal brain growth [53]. EC is particularly effective in single-suture craniosynostosis in infants [22]. Early intervention using EC was reported to reduce the likelihood of requiring a second surgery, which is associated with the natural growth of the brain and dura during skull reshaping [22,43]. After the surgery, helmet therapy is often recommended to guide the skull into a more typical shape as it grows [54]. The key features of this approach include minimal scarring, reduced blood loss, shorter operative time, and quicker recovery compared to traditional open surgical methods [51].

4.1.4. Spring-Assisted Surgery

SAS involves the insertion of specially designed metal springs between sections of the skull bones to gradually expand intracranial volume and correct skull deformities over time. This technique is particularly indicated for mild forms of craniosynostosis, such as sagittal synostosis, where the primary goal is to allow for gradual cranial expansion without extensive surgical remodeling [27,34,46]. The springs are placed through small incisions and exert continuous outward pressure on the skull, promoting bone growth and shape correction [55]. Several months post-surgery, a second procedure is usually needed to remove the springs. In comparison with CVR, SAS is minimally invasive and is associated with reduced surgical time and shorter hospital stays [52]. Progressive skull reshaping is achieved without significant disruption to surrounding tissues. Outcomes are generally positive, with substantial improvements in head shape and intracranial volume and a low risk of complications [34,56].

4.1.5. Strip Craniectomy with Helmet

SC is a surgical method where a narrow strip of bone is removed along the fused suture, allowing for the release of the abnormal cranial growth pattern. The procedure is minimally invasive and is most effective when performed on infants with single-suture craniosynostosis [46,54]. Following the surgery, the infant wears a custom-fitted cranial helmet that gently guides the growth of the skull to a more typical shape. The helmet is worn for several months to ensure optimal reshaping as the skull grows [38]. This approach has the advantage of being less invasive than full cranial vault reconstruction, with a faster recovery time and less risk of significant blood loss [38,46,54]. Outcomes generally include an effective correction of head shape and symmetry, particularly when surgery is performed early, with minimal need for further surgical intervention [57].

4.1.6. Pi Craniectomy

PiC is a specialized technique designed for the treatment of sagittal synostosis. The procedure involves the removal of fused sutures and the reshaping of the skull in a pattern resembling the Greek letter “π” (pi), which allows for significant contouring and expansion of the cranial vault [35]. This approach focuses on achieving a more natural head shape by selectively removing and reshaping parts of the skull, rather than performing a full reconstruction [58]. In sagittal craniosynostosis patients presenting with occipital deformity, occipital remodeling modification during PiC is recommended to increase the stability of the improvement in the angle of the inferior occiput [58].

4.1.7. Pi Plasty

PiP is a modification of Pi craniectomy that emphasizes expanding the fronto-orbital area of the skull. The technique is effective for conditions like metopic synostosis or anterior plagiocephaly [59,60]. This technique involves creating a more extensive reshaping of the cranial bones to increase space in the anterior part of the skull, thereby alleviating any restriction caused by premature suture fusion [39]. The procedure is designed to correct forehead asymmetry and restore a more typical skull shape [61]. PiP has been reported to improve cranial volume with cosmetic enhancement of the forehead and orbital areas [62].

4.1.8. Renier’s “H” Technique

RH is an open cranial vault remodeling procedure used primarily in complex cases of craniosynostosis involving multiple sutures. The procedure entails making an “H”-shaped incision across the skull to facilitate the maximum expansion of intracranial volume and enable the symmetrical reshaping of the head [47]. The “H” incision allows for better access and more extensive remodeling of the skull compared to other techniques, making it suitable for severe or multi-suture synostosis cases [63]. In Slovenia, RH was observed to be ineffective in correcting frontal protrusions, as the results were aesthetically insufficient [64]. In remodeling frontal bossing, RH was found to yield lower CI but successfully increase sagittal length [37].

4.2. Comparisons of the Surgical Techniques

The optimal surgical approach for craniosynostosis remains variable, as numerous techniques have been proposed. This study introduces the novelty of applying Bayesian network meta-analysis to compare multiple surgical methods simultaneously, which provides a more comprehensive assessment than traditional pair-wise comparisons. In this study, we have successfully compared CVR with other approaches, namely EC, ES, PiC, PiP, RH, SAS, and SC, through Bayesian network meta-analysis using published data from nine different studies [20,21,33,34,35,36,37,38,39]. We found that PiP is superior as compared to others, followed by EC, which has the second highest likelihood of yielding superior cephalic index improvement. In comparison with CVR, other techniques, namely EC (SMD = 0.23 [95%CI: −5.47 to 5.63]), ES (SMD = −0.59 [95%CI:−6.07 to 4.94]), PiC (SMD = −1.16 [95%CI:−8.89 to 6.35]), PiP (SMD = −0.07 [95%CI:−9.27 to 8.79]), RH (SMD = −0.96 [95%CI: −6.62 to 4.53]), SAS (SMD = −0.86 [95%CI: −8.25 to 6.18]), and SC (SMD = −1.79 [95%CI: −9.05 to 5.28]), are not significantly more effective. This is in line with the individual studies reporting comparisons between CVR and ES, where the cephalic index between the two groups was not significantly different post-surgery [20,21].
In this study, PiP was revealed to have the highest probability of being associated with higher cephalic index improvement. In one study, PiP was also shown to have similar efficacy in improving the cephalic index as compared to other open approaches (such as parietal reshaping, geometric expansion, parietal–occipital switch, and clamshell craniotomy) [33]. However, in comparison with EC, one study suggested that PiP was less efficacious, particularly after a longer follow-up period [35]. In a study with a 2-year follow-up period, improvements in cranial dimensions were similar among patients receiving PiP, ES, and CVR [65]. Despite having similar efficacy, these techniques vary significantly in terms of medical cost, which requires further studies in the future [66].
ES is a minimally invasive method that utilizes an endoscope on the affected suture(s) in craniosynostosis cases. The technique requires orthotic helmets to aid in postoperative skull reshaping and to optimize the expansion of the brain within the newly released cranial space [21,34]. As for PiP, as an open procedure, it involves removing a segment of the bone located directly behind coronal sutures and on both sides of a sagittal suture. The cranium’s anterior–posterior dimension is reduced, resulting in the bulging of the brain and osseous segments into the biparietal region [35]. The procedure uses midline craniotomy of the fused sagittal suture and paramedian sutures to allow for anterior–posterior shortening and skull widening [33,35].
Surveys among craniofacial surgeons regarding the use of surgical techniques revealed that their preference in choosing the surgical technique is thought to be associated with skull maturation [15]. In patients older than 12 months, a lower skull malleability and higher bone thickness are expected, contributing to the difficulty and reduced efficacy of the surgical procedure [67]. Similarly, in the case of the Pi procedure, it is commonly performed on patients younger than 6 months old [68]. However, when patients are older than 12 months, surgeries with a more invasive approach, such as parietal craniotomy remodeling and parietal distraction osteogenesis, are preferred [33]. In fact, in two different studies included herein, patients undergoing CVR were observed to be older than those undergoing ES or PiC [20,33]. Another study also reported a younger mean age in patients receiving ES as compared to CVR [51].
CVR offers more extensive anatomic exposure to correct the deformity in craniosynostosis cases as compared to ES or EC. This factor is frequently believed to be associated with overcorrection in CVR patients, in whom it was found to be higher than in those receiving endoscopy-assisted surgeries [20]. It is worth noting that, though this is still debatable, synostosis could be induced in other sutures as a result of the surgery [20,65,69]. The use of orthotic helmets following endoscopic surgery might also result in adverse effects, including patient intolerance and skin irritation. Hence, careful consideration by the surgeon based on the age of the patient, severity of the condition, and the side effects and benefits of the surgery is required. This further implies the necessity to assess the long-term effect of the surgery, especially with regard to cranial growth.

4.3. Recommendations

Our findings indicate that PiP and EC are effective in improving the cephalic index in craniosynostosis cases, particularly in younger patients under 12 months, in whom skull malleability is higher. In older patients with less flexible skulls, more invasive approaches like CVR may be necessary. While CVR offers extensive anatomical correction, it presents a higher risk of overcorrection, so it should be reserved for complex cases in which less invasive options may not suffice [20]. Postoperative helmet therapy is essential following ES or EC to support skull reshaping and optimize cranial growth, but it requires careful monitoring to manage discomfort and skin irritation [23,54,66]. Minimally invasive techniques like EC not only reduce blood loss and hospital stays but also lower overall healthcare costs due to fewer complications [70]. Long-term follow-up is crucial to assess cranial growth, cephalic index changes, and potential complications, as variability in skull development may influence outcomes [71].
Future research should address outcomes beyond head shape, such as neurodevelopmental progress and quality of life [18,72]. Studies suggest that earlier surgical interventions correlate with better cognitive and psychosocial development [20,33,73]. Additionally, quality of life, encompassing emotional and social well-being, is increasingly viewed as a key measure of long-term success [18,72]. The experience of the surgical team and hospital resources also affect outcomes; specialized craniofacial teams with adequate resources tend to achieve better results, underscoring the importance of specialized care for these complex cases [74,75].

4.4. Limitations

Our meta-analysis has some limitations. We were unable to retrieve three full-text articles, which might have resulted in selection bias. Additionally, the number of available studies on this topic remains small, limiting the overall sample size and potentially impacting the generalizability of our findings. Disease severity and baseline cephalic index values were relatively consistent across the included studies, which helped minimize bias from these variables. However, patients’ gender and age, particularly the latter, could have resulted in bias in the comparative estimation.
Variability in patient age, especially in surgical interventions, can influence skull malleability and thus affect outcomes, highlighting the need for age-specific approaches in future research. Additionally, long-term follow-up data are limited, especially for minimally invasive techniques, which constrains our understanding of their sustained impact on cranial shape, neurodevelopmental progress, and quality of life. Lastly, the selection of surgical technique is influenced not only by its efficacy but also by patient age, disease severity, potential side effects, and the experience of the surgical team. These factors should be considered in future studies to better understand how they influence surgical success and long-term outcomes.

5. Conclusions

Our meta-analysis suggests that endoscopy-assisted techniques are as efficacious as CVR. PiP, an open localized surgical technique, is more likely to have higher efficacy in improving the cephalic index compared to other techniques. CVR, as it exposes more surgical sites, facilitates precise positioning and maneuvering during the surgery. Despite the increased cephalic index, overcorrection is more frequent in CVR as compared to endoscopic surgeries. The findings of the present study could be used as a basis for clinical decision making during the management of craniosynostosis. Furthermore, this study gives insight into resource allocation to promote the performance of endoscopy-assisted surgery (i.e., through training surgeons and enriching hospital facilities) whilst considering other aspects of the patients. It is essential to carry long-term assessment on the impact of these surgeries, as there are indications for synostosis occurring in other sutures. Overcorrection rates should also be considered in future studies, and evaluating the associated appearance among laypersons is necessary.

Author Contributions

Conceptualization, M.I.A.P. and M.A.; methodology, M.I.; software, M.I.; validation, M.A., B.B., S.A. and S.T.A.-G.; formal analysis, M.I.A.P. and M.I.; investigation, M.I.A.P., S.A. and S.T.A.-G.; writing—original draft preparation, M.I.A.P., M.I., S.A. and S.T.A.-G.; writing—review and editing, M.A. and B.B.; visualization, M.I.; supervision, B.B. All authors have read and agreed to the published version of the manuscript.

Funding

This research received no external funding.

Conflicts of Interest

The authors declare no conflicts of interest.

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Surgeries 05 00078 g001
Figure 1. PRISNMA flow diagram illustrating selection of eligible studies on craniosynostosis surgeries. CI, cephalic index.
Figure 1. PRISNMA flow diagram illustrating selection of eligible studies on craniosynostosis surgeries. CI, cephalic index.
Surgeries 05 00078 g001
Surgeries 05 00078 g002
Figure 2. Network graph for the comparison of craniosynostosis surgeries. CVR, open total cranial vault reconstruction; ES, endoscopic suturectomy; EC, endoscopy-assisted craniectomy; SAS, spring-assisted surgery; SC, strip craniectomy with helmet; PiC, Pi craniectomy; PiP, Pi plasty; RH, Renier’s “H” technique.
Figure 2. Network graph for the comparison of craniosynostosis surgeries. CVR, open total cranial vault reconstruction; ES, endoscopic suturectomy; EC, endoscopy-assisted craniectomy; SAS, spring-assisted surgery; SC, strip craniectomy with helmet; PiC, Pi craniectomy; PiP, Pi plasty; RH, Renier’s “H” technique.
Surgeries 05 00078 g002
Surgeries 05 00078 g003
Figure 3. Forest plot for network pooled estimates of craniosynostosis as compared with CVR. CVR, open total cranial vault reconstruction; ES, endoscopic suturectomy; EC, endoscopy-assisted craniectomy; SAS, spring-assisted surgery; SC, strip craniectomy with helmet; PiC, Pi craniectomy; PiP, Pi plasty; RH, Renier’s “H” technique.
Figure 3. Forest plot for network pooled estimates of craniosynostosis as compared with CVR. CVR, open total cranial vault reconstruction; ES, endoscopic suturectomy; EC, endoscopy-assisted craniectomy; SAS, spring-assisted surgery; SC, strip craniectomy with helmet; PiC, Pi craniectomy; PiP, Pi plasty; RH, Renier’s “H” technique.
Surgeries 05 00078 g003
Table 1. Keyword combinations used in different databases.
Table 1. Keyword combinations used in different databases.
DatabaseKeyword CombinationHits
PubMedallintitle: craniosynostosis cranioplasty OR suturectomy OR
craniectomy OR remodeling OR reconstructive OR reconstruction OR distraction OR osteogenesis		441
ScopusTITLE ((craniosynostos * OR synostos * OR scaphocephaly OR (skull AND anomaly)) AND (cranioplasty OR suturectomy OR craniectomy OR remodeling OR reconstructi * OR (distraction AND osteogenesis)))471
WoS(craniosynostosis OR synostosis OR scaphocephaly OR (skull anomaly)) AND (cranioplasty OR suturectomy OR craniectomy OR remodeling OR reconstructive OR reconstruction OR
(distraction osteogenesis))		460
Scilit(craniosynostosis OR synostosis OR scaphocephaly OR (skull anomaly)) AND (cranioplasty OR suturectomy OR craniectomy OR remodeling OR reconstructive OR reconstruction OR
(distraction osteogenesis))		101
Table 2. Characteristics and outcomes reported in the included studies.
Table 2. Characteristics and outcomes reported in the included studies.
Author, Year [Ref.]Surgical
Technique		CharacteristicsAffected SutureSyndromic StatusCephalic Index
nAge (Month)Gender (M/F)
Al-Shaqsi et al., 2021 [20]CVR3713.4 ± 19.230/7SagittalMix79.3 ± 10
ES182.9 ± 2.814/23SagittalMix74.1 ± 4.4
Albuz et al., 2024 [21]CVR24NRNRMixNo76.2 ± 1.5
EC61NRNRMixNo76.5 ± 1.5
Skolnick et al., 2020 [34]SAS274.5 ± 1.323/4SagittalNo74.3 ± 4.2
EC403 ± 0.929/11SagittalNo77 ± 3.8
Magge et al., 2019 [35]PiC215.1 ± 2.7NRSagittalNo0.7 ± 0.0 a
EC303.1 ± 1.2NRSagittalNo0.8 ± 0.0 a
Taylor et al., 2011 [36]SAS76.3 ± 0.7NRNRNR78.9 ± 1.8
SC75.6 ± 0.5NRNRNR78 ± 4
Spazzapan et al., 2024 [37]RH285.1 ± 1NRSagittalNo72.9 ± 1.5
CVR30NRSagittalNo74.4 ± 1.5
Windh et al., 2008 [39]SAS203.5 ± 0.618/2SagittalNo70.6 ± 3.7
PiP207.1 ± 1.416/4SagittalNo73.4 ± 3.3
Crofts et al., 2023 [38]SC134.6 ± 1.38/5SagittalNR0.7 ± 0.0 a
EC383.1 ± 0.228/10SagittalNR0.8 ± 0.0 a
Chi et al., 2022 [33]CVR3731.2 ± 17.035/8SagittalNo74.6 ± 5.4
PiC66.5 ± 2.2SagittalNo73.9 ± 5.2
a Change in cephalic index; CVR, open total cranial vault reconstruction; ES, endoscopic suturectomy; EC, endoscopy-assisted craniectomy; M/F, male/female; NR, not reported; SAS, spring-assisted surgery; SC, strip craniectomy with helmet; PiC, Pi craniectomy; PiP, Pi plasty; RH, Renier’s “H” technique.
Table 3. Results of critical appraisal using NOS.
Table 3. Results of critical appraisal using NOS.
Author, Year [Ref.]Study DesignSelectionComparabilityOutcomeTotal ScoreRemark
Al-Shaqsi et al., 2021 [20]Cross-sectional★★-★★★5Medium risk
Albuz et al., 2024 [21]Cross-sectional★★★★★★★7Low risk
Skolnick et al., 2020 [34]Cohort★★★★★★★7Low risk
Magge et al., 2019 [35]Cross-sectional★★★★★★★7Low risk
Taylor et al., 2011 [36]Case–control★★★★★★6Medium risk
Spazzapan et al., 2024 [37]Cohort★★★★★★★★8Low risk
Windh et al., 2008 [39]Cohort★★★★★★★★8Low risk
Crofts et al., 2023 [38]Cohort★★★★★★6Medium risk
Chi et al., 2022 [33]Cohort★★★★★6Medium risk
(★), indicate the score; (-), no score.
Table 4. Results of network meta-analysis for the comparison of craniosynostosis surgeries with pooled estimates presented in SDM (95%CI).
Table 4. Results of network meta-analysis for the comparison of craniosynostosis surgeries with pooled estimates presented in SDM (95%CI).
CVR0.23 (−5.47–5.63)−0.59 (−6.07 to 4.94)−1.16 (−8.89 to 6.35)−0.07 (−9.27 to 8.79)−0.96 (−6.62 to 4.53)−0.86 (−8.25 to 6.18)−1.79 (−9.05 to 5.28)
EC−0.79 (−8.57 to 7.19)−1.36 (−6.85 to 4.20)−0.31 (−7.29 to 7.08)−1.20 (−9.14 to 6.88)−1.09 (−5.54 to 3.54)−2.01 (−6.46 to 2.65)
ES−0.58 (−10.13 to 9.05)0.54 (−10.08 to 11.19)−0.41 (−8.31 to 7.35)−0.28 (−9.35 to 8.80)−1.2 (−10.58 to 7.94)
PiC1.07 (−7.84 to 10.31)0.19 (−9.20 to 9.66)0.26 (−6.66 to 7.41)−0.65 (−7.67 to 6.44)
PiP−0.89 (−11.82 to 9.77)−0.80 (−6.38 to 4.84)−1.70 (−8.87 to 5.47)
RH0.09 (−9.04 to 9.44)−0.82 (−9.82 to 8.42)
SAS−0.882 (−5.26 to 3.75)
SC
Data are presented as SMD (95%CI). Each cell shows the effect of the column-defining intervention relative to the row-defining intervention. CVR, open total cranial vault reconstruction; ES, endoscopic suturectomy; EC, endoscopy-assisted craniectomy; SAS, spring-assisted surgery; SC, strip craniectomy with helmet; PiC, Pi craniectomy; PiP, Pi plasty; RH, Renier’s “H” technique.
Table 5. Rank probabilities of craniosynostosis surgeries based on network Bayesian meta-analysis.
Table 5. Rank probabilities of craniosynostosis surgeries based on network Bayesian meta-analysis.
Surgical TechniquesRank 1Rank 2Rank 3Rank 4Rank 5Rank 6Rank 7Rank 8
CVR0.1420.2290.2010.1360.1180.1010.0590.015
EC0.1950.2460.2040.1750.1020.0530.0220.004
ES0.1500.1090.1170.1290.1230.1160.1260.131
PiC0.0820.0770.0830.1090.1410.1380.1860.184
PiP0.2730.1280.1210.1130.0960.0930.0810.097
RH0.1040.0860.1010.1050.1190.1460.1520.186
SAS0.0290.0880.1180.1550.1870.2080.1570.058
SC0.0260.0370.0560.0790.1150.1460.2170.326
CVR, open total cranial vault reconstruction; ES, endoscopic suturectomy; EC, endoscopy-assisted craniectomy; SAS, spring-assisted surgery; SC, strip craniectomy with helmet; PiC, Pi craniectomy; PiP, Pi plasty; RH, Renier’s “H” technique.
Table 6. Summaries of the surgical techniques comparatively analyzed in the network meta-analysis.
Table 6. Summaries of the surgical techniques comparatively analyzed in the network meta-analysis.
SurgeryKey FeaturesIndicationRelated FindingsRef.
Open total cranial vault reconstructionThe procedure includes modifying and restructuring the bones of the skull to enlarge the intracranial space and fix the shape of the head.Used for complicated or multi-suture craniosynostosisExtensive skull remodeling, longer recovery time, but results in a comprehensive correction[41]
Endoscopic suturectomySmall, minimally invasive incisions are performed to remove fused sutures, which are frequently followed by helmet treatment to reshape the skull.Ideal for infant single-suture craniosynostosisLess scarring and faster healing; limited usage in more complicated cases[42]
Endoscopy-assisted craniectomyEndoscopic procedures are used with a minimally invasive approach to remove sections of the skull damaged by premature suture fusion.Synostosis with a single suture performed in infantsHelmet treatment may be required following surgery for reshaping; minor blood loss is expected[43]
Spring-assisted surgeryMetal springs are implanted between the skull bones to progressively expand intracranial volume over time; the springs are then withdrawn.Mild craniosynostosis and sagittal synostosisNon-invasive postoperative repair; progressive skull reshaping[44]
Strip craniectomy with helmetThe procedure involves the removal of fused sutures in a thin strip, followed by helmet treatment to guide skull development.Craniosynostosis with a single suture in infantsReshaping process through helmet use is less invasive compared to full vault reconstruction and occurs gradually over time[44]
Pi craniectomyTo enhance head contour, a reshaping technique is performed to remove fused sutures and redesign the skull in the shape of the Greek letter “π”.Sagittal synostosisCorrects head shape and reduces the need for further procedures[45]
Pi plastyThis procedure is similar to Pi craniectomy but emphasizes creating more space in the fronto-orbital area and restoring normal skull shape.Metopic synostosis or anterior plagiocephalyEffective for contouring and expanding the frontal area, particularly in cases with anterior cranial deformities[46]
Renier’s “H” techniqueThe procedure of cranial vault reconstruction includes creating an incision in the shape of an “H” to enable the maximum expansion of the skull and achieve symmetrical reshaping.Used in more complicated cases of craniosynostosisMultiple-suture synostosis; symmetrical skull reshaping and expansion[47]
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Putra, M.I.A.; Amirsyah, M.; Budiman, B.; Amirah, S.; Al-Gunaid, S.T.; Iqhrammullah, M. Comparison of Surgical Techniques in Managing Craniosynostosis: Systematic Review and Bayesian Network Meta-Analysis. Surgeries 2024, 5, 970-985. https://doi.org/10.3390/surgeries5040078

AMA Style

Putra MIA, Amirsyah M, Budiman B, Amirah S, Al-Gunaid ST, Iqhrammullah M. Comparison of Surgical Techniques in Managing Craniosynostosis: Systematic Review and Bayesian Network Meta-Analysis. Surgeries. 2024; 5(4):970-985. https://doi.org/10.3390/surgeries5040078

Chicago/Turabian Style

Putra, Muhammad Ikhlas Abdian, Mirnasari Amirsyah, Budiman Budiman, Shakira Amirah, Seba Talat Al-Gunaid, and Muhammad Iqhrammullah. 2024. "Comparison of Surgical Techniques in Managing Craniosynostosis: Systematic Review and Bayesian Network Meta-Analysis" Surgeries 5, no. 4: 970-985. https://doi.org/10.3390/surgeries5040078

APA Style

Putra, M. I. A., Amirsyah, M., Budiman, B., Amirah, S., Al-Gunaid, S. T., & Iqhrammullah, M. (2024). Comparison of Surgical Techniques in Managing Craniosynostosis: Systematic Review and Bayesian Network Meta-Analysis. Surgeries, 5(4), 970-985. https://doi.org/10.3390/surgeries5040078

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