Shared decision making in the treatment of type B ulnar polydactyly: a systematic review and meta-analysis on parental satisfaction and complications

Abstract

Introduction:

Type B ulnar polydactyly is a common congenital hand anomaly. Treatment options include suture ligation, vascular clip ligation and excision under local or general anaesthesia. However, the optimal treatment remains unclear, and evidence on parental perspectives is limited. This review investigated treatment outcomes for type B ulnar polydactyly, in combination with parental satisfaction and preferences.

Methods:

A systematic search of Embase, Pubmed, Medline, Web of Science and Cochrane Central Register of Controlled Trials was conducted up to March 2025 for studies reporting parental satisfaction with treatment outcome or aesthetic appearance, parental prioritizing factors or complications. Meta-analyses were performed on pooled parent-reported satisfaction scores and complication data.

Results:

Fourteen studies, comprising 885 patients with 1279 supernumerary digits, met the inclusion criteria. Overall, parental satisfaction was high across all treatments. Excision under local anaesthesia resulted in significantly greater satisfaction on both treatment outcome and aesthetic appearance compared with suture ligation and excision under general anaesthesia. No parent-reported data were available on vascular clip ligation. Parents choosing suture ligation or excision under local anaesthesia mentioned avoiding anaesthesia risks and quick treatment as priorities, while parents selecting excision under general anaesthesia prioritized pain avoidance. The pooled complication rate was lowest for excision under local anaesthesia (0.01) and highest for suture ligation (0.16).

Conclusion:

Excision under local anaesthesia showed the best results regarding treatment of type B ulnar polydactyly, with the highest parental satisfaction outcomes and lowest complication rates. However, high parental satisfaction was observed in all treatment modalities and differences in outcomes were small. Parents primarily based their treatment choice on avoiding risks (with ligation or local anaesthesia) or avoiding pain (with general anaesthesia). Our results can be used for effective decision making.

Keywords

Decision making patient reported outcome measures polydactyly postoperative complications surgical procedures treatment outcomes

Introduction

Polydactyly is the most common congenital anomaly of the hand and foot (Bjorklund and O’Brien, 2022). Ulnar polydactyly, defined by the presence of a rudimentary digit along the fifth ray, is the most frequent type, with an incidence estimated at 1–2 per 1000 live births (Zhou et al., 2004). Extra digits are classified by Temtamy and McKusick (1978) as either type A or type B.

Despite the current literature on interventions for type B ulnar polydactyly, controversy persists regarding the optimal approach, as all have advantages and disadvantages (Chopan et al., 2020; Samarendra et al., 2022). Extra digits can be removed by ligation using a suture or vascular clip at the base or by surgical excision under local or general anaesthesia (Figure 1). Ligation can be performed shortly after birth, but is only possible when the stalk is narrow (Mullick and Borschel, 2010). Surgical excision is suitable for all type B variants. However, anaesthesia-related risks delay the procedure until an older age (Davidson and Vutskits, 2017; Houck and Vinson, 2017), which may cause emotional stress for parents awaiting treatment (Samra et al., 2016). Excision under local anaesthesia enables earlier intervention, but requires patient and parent cooperation, experienced staff and adequate facilities (Samra et al., 2016). The most optimal age is up to 3 months, as increased awareness in older children makes this intervention more challenging (Gajewska et al., 2013).

Figure 1.

Intervention methods for treatment of type B ulnar polydactyly: (a) suture ligation; (b) vascular clip ligation using multiple clips. The digit is allowed to necrose or is excised, and (c) surgical excision via elliptical incision enabling proximal resection.

Despite these considerations, comparative evidence remains limited, as is the case for parental satisfaction with treatment results, factors influencing parental decision making and motivations for treatment choice. As patients are too young to make the decision themselves, parental involvement and cooperation are essential and may contribute to achieving a favourable result (Haine-Schlagel and Walsh, 2015; Mackie et al., 2018; Schreuder et al., 2019). Therefore, this review evaluates treatment outcomes of type B ulnar polydactyly focusing on parental satisfaction and decision making. Additionally, the complication rate per intervention will be explored and compared.

Methods

A systematic review of the literature on parental outcomes and complications of treatment of type B ulnar polydactyly was developed in accordance with the Cochrane Handbook for Systematic Reviews of Interventions (Higgins et al., 2024) and the PRISMA guidelines (Moher et al., 2009).

Literature search

In collaboration with a medical information specialist, a systematic search strategy was developed. The complete search strategy is provided in Supplementary file S1. The initial searches were conducted in Embase, PubMed and Medline on 9 January 2025. The search was repeated on 12 March 2025 to ensure that the most recent studies were included in review. The databases that were searched in addition included Web of Science, Cochrane Central Register of Controlled Trials and Google Scholar. Beyond database searches, a forward and backward citation search was conducted for all included full-text studies to identify further potentially relevant studies.

Study selection

Two authors (SHB and NJN) independently screened studies for eligibility based on the title and abstract. Studies were included if participants had type B ulnar polydactyly and underwent ligation (suture and/or clip) or surgical excision (under local or general anaesthesia), and if parental outcomes on satisfaction with treatment outcome, satisfaction with aesthetic appearance or complications related to treatment were reported. Studies were excluded if they only focused on radial or central polydactyly, polydactyly of the feet, alternative interventions or unrelated outcomes, and if most participants in the study already had received treatment for type B ulnar polydactyly. Furthermore, reviews, case series with fewer than five patients, case reports, descriptive studies, animal studies, conference abstracts, poster presentations, studies written in another language than Dutch or English and non-full-text articles were excluded. After selection, the senior author (ESS) was consulted in case of conflicts.

Data extraction and quality scoring

Data were extracted from the selected studies using a standardized collection form. The collected variables included year of publication, study type, level of evidence, number of patients and type B ulnar polydactyly treated, sex, age at intervention, ethnic background, family history of ulnar polydactyly, type of technique, outcome measurements and follow-up time.

The primary endpoints were parent-reported satisfaction with treatment outcome and satisfaction with aesthetic appearance. Satisfaction with treatment outcome referred to overall satisfaction with the treatment process and need for revision surgery. Satisfaction with aesthetic appearance referred to the visual appearance of the hand and/or scar. Both parental outcomes were assessed through satisfaction questionnaires.

The secondary endpoints represented prioritizing factors in parental decision making and short-term and long-term complications. Prioritizing factors referred to parent-reported motivations that influenced their decision making regarding treatment of ulnar polydactyly type B. Short-term complications included bleeding, necrosis, infection and failure to auto-amputate. Long-term complications included clinically relevant neuroma, presence of a residual bump, and re-operations.

The various therapies were placed in four different groups for synthesis: ligation via sutures, ligation via clips, surgical excision under local anaesthesia and surgical excision under general anaesthesia. Quality assessment was performed using the study quality assessment tools of the National Institutes of Health (National Heart, Lung, and Blood Institute, 2014), for case series and retro- and prospective cohort studies (Figure S2). The strength of evidence of each study was assessed using the classification of strength of evidence by Jovell and Navarro-Rubio (1995).

Reported age at the time of procedure and follow-up period were standardized by converting to weeks and months, and values were estimated as median, mean and range to ensure comparability across studies. The included studies used heterogenous scales for parental outcomes, limiting the compatibility of data. One study (Singer et al., 2014) used a visual analogue scale to report satisfaction with the postoperative results. Five- and 10-point Likert scales were used to report satisfaction with treatment outcome or aesthetic appearance. To evaluate parental outcomes, satisfaction scores were rescaled to a standardized 10-point scale, with 10 indicating maximal satisfaction and 1 indicating minimal satisfaction. Median values and ranges of satisfaction scores were derived from reported results or from raw data obtained from Goebel et al. (2022) when published data were incomplete or unsuitable for statistical analysis.

Statistical analysis

Parental scores regarding satisfaction with treatment outcome and satisfaction with aesthetic appearance were pooled per intervention group. As the individual satisfaction data were not normally distributed, the Kruskall–Wallis H-test was conducted to assess differences across interventions for each outcome. Where significant differences were found, Dunn’s post hoc test with Holm correction was used for pairwise comparisons.

The complication rate per all treated supernumerary digits per intervention was visualized using a forest plot. Standard deviations and confidence intervals were calculated when not reported. The weight of individual studies was computed based on the within-study variance and the estimated between-study variance for the corresponding intervention subgroup. The overall effect was evaluated using the random effects model. All analyses were performed using R (R Core Team, 2021).

Results

The total literature search yielded 1526 publications. After title and abstract screening and removal of duplicates, 62 studies were assessed in full text and led to inclusion of 14 studies, which comprised seven observational cohort studies (Bjorklund and O’Brien, 2022; Ganju et al., 2024; Goebel et al., 2022, 2023; Mosa et al., 2024; Rayan and Frey, 2001; Samra et al., 2016) and seven case series (Carpenter et al., 2016; Katz and Linder, 2011; Mills et al., 2014; Parwez Sajad Khan, 2011; Singer et al., 2014; Stults and Peljovich, 2023; Watson and Hennrikus, 1997), as presented in the PRISMA flowchart (Figure 2). These studies reported 885 patients having 1279 supernumerary digits. Age at intervention ranged from 2 days to 10 years old. The reported follow-up period ranged from 1 week to 14 years (Table 1). Quality assessment classified eight studies as fair, three as good and three as poor (Figure S2).

Figure 2.

PRISMA flow chart.

Table 1.

Overview of study and patient characteristics by intervention.

Author and year	Study design	LoE	Number of digits (number of patients)	Number of bilateral patients (%)	Age at procedure¹	Male (%)	African/American (%)	Follow-up¹
Suture ligation
Watson and Hennrikus (1997)	Prospective	VIII	37 (21)	16 (76)	Newborns	12 (57)	14 (67)	20 (12–37)
Ganju et al. (2024²)	Retrospective	VI	66 (57)	NA	2 (0–14)	31 (54)	36 (67)	96
Vascular clip ligation
Mills et al. (2014)	Retrospective	VIII	231 (132)	94 (71)	8 (2–40)	77 (58)	81 (61)	⩾ 24
Excision under local anaesthesia
Katz and Linder (2011)	Prospective	VIII	15 (11)	4 (36)	2 or 3 days	6 (45)	NA	12 (NA)
Carpenter et al. (2016)	Retrospective	VIII	38 (26)	12 (46)	14 (1–219)	NA	NA	NA
Bjorklund and O’Brien (2022)	Retrospective	VI	82 (51)	31 (61)	11 (7–14)	31 (61)	33 (65)	1 week (NA)
Stults and Peljovich (2023)	Retrospective	VIII	78 (48)	30 (63)	10 (4–28)	30 (62)	64 (75)	38 (6–97)
Ganju et al. (2024²)	Retrospective	VI	192 (101)	NA	10 (0–33)	54 (53)	70 (67)	94
Mosa et al. (2024³)	Retrospective	VI	38 (22)	16 (73)	2 (0–11)	17 (77)	NA	NA
Excision under general anaesthesia
Parwez Sajad Khan (2011)	Prospective	VIII	20 (10)	10 (100)	79 (26–157)	5 (50)	NA	24 (NA)
Singer et al. (2014)	Retrospective	VIII	53 (32)	20 (63)	37 (0–522)	20 (63)	NA	59 (25–120)
Bjorklund and O’Brien (2022)	Retrospective	VI	67 (40)	27 (67)	17 (13–25)	24 (60)	22 (55)	NA
Mosa et al. (2024³)	Retrospective	VI	59 (33)	26 (79)	11 (0–66)	18 (55)	NA	NA
Suture ligation and excision under general anaesthesia
Rayan and Frey (2001⁴)	Retrospective	VI	139 (148)	NA	NA	57	103 (70)	NA
Goebel et al. (2022)	Retrospective	VI	69 (69)	0 (0)	43 (NA)	NA	45 (65)	133 (103–163)
Goebel et al. (2023)	Retrospective	VI	70 (70)	0 (0)	NA	NA	47 (67)	27 (2–60)
Suture ligation, excision under local and general anaesthesia
Samra et al. (2016)	Prospective	VI	25 (14)	11 (79)	Newborns	NA	12 (86)	3

LoE: Level of evidence; NA, not available.

Age (weeks) and follow-up (months) presented as mean (range) unless otherwise specified.

Median follow-up estimated from reported data.

Age at referral.

Treated digits reconstructed from author correspondence.

Parental outcomes

Eight studies (Bjorklund and O’Brien, 2022; Ganju et al., 2024; Goebel et al., 2022, 2023; Katz and Linder, 2011; Samra et al., 2016; Singer et al., 2014; Stults and Peljovich, 2023) evaluated parental outcomes covering 234 parent sets regarding satisfaction with treatment outcome and/or aesthetic appearance. The study by Mills et al. was the only one included on vascular clip ligation and did not report any parental outcomes. Therefore, parental outcomes were not available for vascular clip ligation. Across every intervention, studies reported high satisfaction scores for treatment outcome and aesthetic appearance, indicating great parental contentment regarding each intervention (Table 2).

Table 2.

Overview of parental outcomes of individual studies by intervention.

Author and year	Number of parent sets	Treatment outcome¹	Aesthetic appearance¹	Follow-up¹
Suture ligation
Samra et al. (016²)	1	10	10	3
Goebel et al. (2022)	24	10 (1–10)	10 (1–10)	133 (58–208)
Goebel et al. (2023²)	28	9.8	9.4	27 (2–60)
Ganju et al. (2024^2,3)	41	NA	NA	96
Excision under local anaesthesia
Katz and Linder (2011²)	11	NA	NA	12 (NA)
Samra et al. (2016²)	10	9.8	9.7	3
Bjorklund and O’Brien (2022⁴)	17	10 (8–10)	NA	1 week (NA)
Stults and Peljovich (2023)	34	10 (10–10)	10 (10–10)	38 (6–97)
Ganju et al. (2024^2,3)	94	NA	NA	94
Excision under general anaesthesia
Singer et al. (2014)	20	10 (1–10)	NA	59 (25–120)
Samra et al. (2016²)	2	10	10	3
Goebel et al. (2022)	45	10 (5–10)	10 (4–10)	133 (58–208)
Goebel et al. (2023²)	42	9.8	9.2	27 (2–60)

NA, not available.

Parental outcomes and follow-up period presented as median (range) and mean (range), respectively.

Outcomes reported as mean or NA owing to binary or unstratified data.

Median follow-up estimated from reported data.

Five-point Likert scale assumed based on wording consistent with similar studies.

Four studies (Bjorklund and O’Brien, 2022; Goebel et al., 2022; Singer et al., 2014; Stults and Peljovich, 2023) provided individual-level data to evaluate parental satisfaction with treatment outcome and aesthetic appearance, which enabled direct comparison. Studies were pooled for each intervention group, and data included a total of 140 parents sets. Median and IQR were calculated using the combined data within each group (Table 3). The results in Table 3 reveal a high level of satisfaction regarding both parental outcomes across all interventions. Despite extremely high satisfaction scores across all interventions, we found a significant difference between all three interventions for both satisfaction with treatment outcome (p = 0.009) and aesthetic appearance (p = 0.004). Excision under local anaesthesia appeared to result in the most favourable outcomes for both parental outcomes (Figure 3).

Table 3.

Overview of pooled parental satisfaction outcomes by intervention.

Intervention	Number of parent sets	Treatment outcome	Aesthetic appearance
Suture ligation	24	10.0 (7.5–10)	10.0 (8.5–10)
Excision under local anaesthesia	51	10.0 (10–10)	10.0 (10–10)
Excision under general anaesthesia	65	10.0 (10–10)	10.0 (10–10)

Figure 3.

Parental satisfaction with treatment outcome (a) and aesthetic appearance (b) following interventions for type B ulnar polydactyly. The horizontal lines represent the median and the boxes represent the IQR. Individual data points show within (blue) and outside (orange) the IQR.

Prioritizing factors

Few studies (Bjorklund and O’Brien, 2022; Goebel et al., 2023; Samra et al., 2016; Stults and Peljovich, 2023) reported parental priorities in treatment decision making (Table 4). Studies only briefly reported reasons for intervention preference, without specifying the exact number of parents and how this information exactly was collected. The following main findings were extracted:

When parents opted for suture ligation or excision under local anaesthesia, parents mainly reported avoiding anaesthesia-related risks as motivational factor.

In all studies regarding excision under local anaesthesia, parents prioritized promptness of treatment.

Studies investigating excision under general anaesthesia revealed avoiding potential pain as the prioritizing factor mostly.

Table 4.

Parental prioritizing factors regarding decision making.

Intervention	Motivational factors
Suture ligation
Samra et al. (2016)	Fear of a complication owing to surgeryAvoiding anaesthesia-related risksConcerns for pain
Goebel et al. (2023)	Promptness of treatmentAvoiding anaesthesia related risksPositive family history
Excision under local anaesthesia
Samra et al. (2016)	Promptness of treatmentAvoiding anaesthesia related risksAvoiding potential pain
Bjorklund and O’Brien (2022)	Promptness of treatmentAvoiding anaesthesia related risks
Stults and Peljovich (2023)	Promptness of treatmentAvoiding anaesthesia related risks
Excision under general anaesthesia
Samra et al. (2016)	Avoiding potential pain
Goebel et al. (2023)	Fear of complications from suture ligationAvoidance of pain at the ligature siteInfluence from their physician

Complications

Complication rates from 12 studies were analysed across all four interventions. Residual nubbins, neuromas and re-operations occurred mostly after suture ligation and vascular clipping. Superficial wound infections occurred mostly after excision under local and general anaesthesia, and several neuromas, nubbins and re-operations were also reported. A meta-analysis assessed pooled complication rates per technique (Figure 4). The lowest complication rate was observed after excision under local anaesthesia (1.0%), followed by excision under general anaesthesia (3.0%) and vascular clipping (16%), while suture ligation showed the highest rate (53%). Subgroup differences were statistically significant (p < 0.001). The overall pooled complication rate was 6.0%.

Figure 4.

Individual and pooled complication rates per intervention. The complication rate was calculated by dividing the number of complications by the number of digits.

Discussion

This systematic review investigates parental satisfaction and complication rates across different treatment methods for type B ulnar polydactyly. Overall, each intervention showed high parental satisfaction with treatment outcome and aesthetic appearance, and most interventions demonstrated low complication rates. When comparing the studies, excision under local anaesthesia seemed to be associated with the most favourable results, owing to the highest satisfaction and lowest complication rates. Owing to insufficient data, these findings do not apply to treatment via vascular clip ligation.

In our review, analysis of individual satisfaction data indicated that excision under local anaesthesia seemed to be associated with the most beneficial results for parent satisfaction. This might be related to the fact that a few parents reported negative scores in the suture ligation and excision under general anaesthesia groups. This could explain the statistically significant differences observed, although overall satisfaction was high across all treatment modalities for both treatment outcome and aesthetic appearance. These findings should therefore be interpreted with clinical relevance in mind, as high overall satisfaction may limit the practical significance of the observed differences. While parental satisfaction is of paramount importance in paediatric surgery, it remains inherently subjective and can be influenced by non-clinical factors such as communication quality, empathy, expectations and cultural norms (Butler et al., 2014; Kuo et al., 2012). Therefore, excision under local anaesthesia should not be the only method being recommended, as parental satisfaction depends on more than clinical results alone. Surgeons should consider which treatment fits best with the preferences and situation of each family and apply shared decision making. Moreover, decision making is not only influenced by parental preferences but also by logistical limitations. For instance, surgeons are sometimes forced to opt for excision under local anaesthesia owing to the absence of a paediatric anaesthesiologist. In addition, the hospital may either lack available operating rooms or a minor treatment centre for excision under local anaesthesia, making ligation with clips or sutures a logistically feasible option. The choice of procedure is, therefore, also determined by the hospital’s available resources and logistical possibilities. Since parental satisfaction data were unavailable for vascular clipping, these findings do not apply for this intervention.

The parental outcomes for vascular clip ligation remain unclear. The only study in this review on clip ligation (Mills et al., 2014) showed relatively high complication rates. The most reported complication included neuroma formation, followed by revision surgery. However, the authors’ experience (CAN and ESS) with complications following vascular clip ligation appears to differ from what is described in the current literature. Nevertheless, in the absence of comparative studies, definitive conclusions regarding the potentially high complication rates associated with vascular clip ligation cannot be drawn. Future studies should investigate parental outcomes and complication rates following vascular clip ligation.

Prioritizing factors in parental decision making have been minimally described in the literature. In our review, we were able to provide an overview of reported motivational factors from four studies (Bjorklund and O’Brien, 2022; Goebel et al., 2023; Samra et al., 2016; Stults and Peljovich, 2023). In addition to the limited evidence on parental outcomes, data on long-term results in children who have reached adulthood are still lacking. Their experience could contribute to improving the treatment of type B ulnar polydactyly. Validated questionnaires are needed to align and compare the data, which were not applied to current studies. Our findings highlight the need for further research on this topic, which could affect shared decision making and better alignment between clinical practice and both parental and patient preferences.

Complication rates were compared by pooling individual studies according to the intervention method. Our forest plot presented the lowest pooled complication rate for excision under local anaesthesia. Suture ligation showed a particularly high complication rate, followed by vascular clip ligation. Most complications comprised neuromas and residual nubbins. This can be explained by the fact that surgical excision allows for proximal transection of the nerve, which is not possible with ligation (Patillo and Rayan, 2011). The difference in complication rates between suture ligation and vascular clip ligation might be due to the mechanism of both methods. Vascular clips presumably stay tightly fixed at the very base of the digits, while sutures tend slip off the base toward the thinnest part of the stalk, which could result in painful residual nubbins (Mills et al., 2014).

There are some restrictions to our analysis. Variation in the definition and reporting of complications across studies could possibly lead to reporting bias and underestimation of true complication rates. Additionally, the variation in follow-up period is an important confounding factor, since complications like clinically relevant neuromas, scar formation and revision surgery are time dependent (Oliveira et al., 2018; Tullington and Gemma, 2023) and could potentially be long-term problems. Therefore, excision under local anaesthesia seems to lead to the most beneficial results, however, the findings of our analysis should be evaluated with these considerations.

This review contains some limitations. Owing to incomplete data, only four studies were included in the meta-analysis of 140 parent satisfaction scores. Our meta-analys is therefore limited by these small subgroup sizes, including one subgroup being represented by a single study (Goebel et al., 2022), and by the use of non-validated questionnaires. Conversion of non-standardized measurement methods, such as five-point Likert scales and visual analogue scales to a 10-point scale required recalculation of some parental satisfaction scores. Consequently, some of the values used in the analysis represent converted rather than exact original data. However, this standardization to a 10-point scale enabled comparison of individual scores across studies, which allowed for meta-analysis. High satisfaction scores induced ceiling effects, limiting the ability to detect differences between groups.

A major strength of our study is its rigorous methodology, including a thorough literature search, transparent reporting and systematic analysis of parental outcomes. Previous reports (Chopan et al., 2020; Samarendra et al., 2022) included no meta-analysis of individual data and could not identify a preferred method. Previous reports also report complication rates but lack accuracy. Samarenda et al. reported similar complication rates across interventions to those found in our analysis, but individual studies (Parwez Sajad Khan, 2011; Watson and Hennrikus, 1997) showed much higher rates, probably owing to bilateral polydactyly. To address this, we reported complication rates per digit for greater clinical accuracy. Additionally, we analysed suture ligation and vascular clip ligation separately for nuanced insights and included follow-up durations to address potential underreporting.

Parents should be informed about both clinical and parental satisfaction outcomes, and their motivational factors should be explored to ensure that the most appropriate treatment is offered for each family.

Supplemental Material

sj-docx-1-jhs-10.1177_17531934261447693 – Supplemental material for Shared decision making in the treatment of type B ulnar polydactyly: a systematic review and meta-analysis on parental satisfaction and complications

Supplemental material, sj-docx-1-jhs-10.1177_17531934261447693 for Shared decision making in the treatment of type B ulnar polydactyly: a systematic review and meta-analysis on parental satisfaction and complications by S.H. ten Berge, N.J. Nieuwdorp, E.B. Burger, C.A. van Nieuwenhoven and E.S. Smits in Journal of Hand Surgery (European Volume)

Footnotes

Acknowledgements

None.

Declaration of conflicting interests

The authors declared no potential conflicts of interest with respect to the research, authorship and/or publication of this article.

Funding

The authors received no financial support for the research, authorship and/or publication of this article.

Ethical approval

Not applicable.

Informed consent

Not applicable.

ORCID iDs

S.H. ten Berge

N.J. Nieuwdorp

E.B. Burger

C.A. van Nieuwenhoven

E.S. Smits

Supplemental material

Supplemental material for this article is available online.

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