Talectomy for the Treatment of Rigid Nonidiopathic Clubfoot Deformity: Long-term Follow-up

Abstract

Background

Clubfoot occurs in 30% of patients with myelomeningocele (MM) and is the most frequent foot deformity in patients with arthrogryposis (ART). Patients have high recurrence rates with more conservative interventions and may require talectomy. This study investigated the long-term clinical outcomes of talectomy in patients with MM and ART and identified factors associated with favorable outcomes.

Methods

A retrospective chart review was performed on cases of clubfoott in MM and ART between 1975 and 2010, excluding if follow-up was <5 years or had incomplete charts. A plantigrade, stable, and braceable foot was graded as a good result. Statistical analysis included descriptive statistics, chi-square with continuity correction, Spearman correlation, and Mann-Whitney, with P ​< ​.05 reaching significance.

Results

In total, 944 cases of clubfoot in patients with MM and ART were identified. Of those, 53 underwent talectomy and 31 feet were included in the analysis. Average age on talectomy date was 4.16 ​± ​2.46 years and the mean follow-up since surgery was 15.71 ​± ​8.43 years. “Good results” were found in 24 (77.4%) feet, while 7 feet (22.6%) required additional surgery. Older age at talectomy date was associated with good results at the last clinical visit (P ​= ​.03). Primary talectomies were associated with more subsequent surgeries per foot when compared with salvage talectomies (P ​= ​.03). A Functional Mobility Scale (FMS) of 3-1-1 or higher was associated with a good outcome (P ​= ​.022) and all “poor results” (7 feet, 22.6%) were found in patients with FMS lower than 3-1-1.

Conclusions

Talectomy was performed in 5.6% of cases, demonstrating its rare use in treatment of clubfoot in patients with MM and ART. Nevertheless, these long-term outcomes demonstrate that talectomy surgery may be a good option for the treatment of nonidiopathic clubfoot. Good outcomes are maintained at long-term follow-up, suggesting a satisfactory correction remained until adulthood. Finally, a higher FMS was a predictor of success, suggesting that this procedure should be performed in ambulatory patients.

Key Concepts

• (1)Talectomy remains a rare procedure, utilized in only 5.6% of clubfoot cases of patients with myelomeningocele or arthrogryposis.
• (2)“Good” results, defined as a plantigrade, stable, and braceable foot was achieved in 77.4% of patients.
• (3)Older age at time of talectomy was associated with improved results.
• (4)A Functional Mobility Scale (FMS) of 3-1-1 or higher was associated with good outcomes and thus may be a predictor of success.

Level of Evidence

Level IV

Introduction

Clubfoot, or talipes equinovarus (TEV), is a common anomaly in children with spina bifida, presenting at birth in at least 30% of patients with myelomeningocele (MM) and is the most frequent foot deformity in patients with arthrogryposis (ART) [[1], [2], [3]]. The treatment of severe clubfoot in MM and ART has evolved from always intervening surgically to first initiating serial casting with the Ponseti method, similar to idiopathic clubfoot [1]. Despite initially favorable results with the Ponseti method in MM and ART, high recurrence rates and need for surgical releases indicates that the treatment of these resistant feet remains a challenge [1,2,4,5]. Recurrence is due to the teratologic nature that markedly differs from idiopathic clubfoot. Structural changes, fibrosis, and stiffness of the soft tissues make it less responsive to conservative treatment and more likely to require surgery [[1], [2], [3]]. When the clubfoot deformity recurs or is too severe to be corrected by soft tissue release only, talectomy may be indicated to achieve a stable, braceable, and plantigrade foot [1,3,4]. In rare cases, primary talectomy may also be considered.

Our institution's experience has shown that talectomy can correct the hindfoot equinus and varus deformities in patients with MM and ART [6]. Although the need for talectomy has decreased in this patient population, its place in modern pediatric foot and ankle surgery remains [7]. However, few studies have reported on the long-term follow-up and clinical results, but limited data indicates satisfactory results ranging from 33% to 79% of feet [4,[8], [9], [10], [11], [12]]. Thus, this retrospective study of patients treated by the senior author (LD) presents the long-term results of talectomy for the treatment of resistant clubfoot deformity in MM and ART.

Materials and methods

After IRB approval, a retrospective chart review was carried out to identify patients with clubfoot treated between 1975 and 2010. Talectomy was performed as a primary or salvage procedure in 53 (5.6%) of 944 feet (625 patients) identified to have neuromuscular clubfoot. Indications for talectomy included failure of conservative treatments (casting); a severe deformity indicating that a soft tissue release only would not provide adequate correction (primary talectomy); rigid deformity recurrence after prior surgical intervention that limited function, inability to tolerate bracing due to skin irritation, and/or decreased ability to stand/ambulate for long periods of time secondary to foot pain/deformity; increased risk of complications in a soft tissue release due to level of deformity, rigidity, or scar tissue from previous surgeries; or a foot in which additional osteotomies would not achieve correction (salvage talectomy). Patients were excluded if follow-up was less than 5 years or for incomplete charts.

Procedures performed (before, concomitant, and after talectomy), complications, deformity recurrence, Function Mobility Scale (FMS), and clinical results at the last visit were also collected. Residual deformities after talectomy were graded as recurrence, regardless of the type or interval at which further deformity was reported. A plantigrade, stable, and braceable foot at final follow-up was graded as “good result.” A residual minor deformity at the last visit was only accepted as “good result” if it did not require further treatment and patients did not report a limit in function (standing or ambulation) secondary to foot pain/deformity or inability to tolerate bracing due to skin irritation. Feet showing moderate or severe deformities were graded as “poor”.

All surgeries were performed by the senior author at a single tertiary care pediatric referral center. The surgical technique used is briefly described here [1]. Through an Ollier incision, the talonavicular, tibiotalar, and subtalar joints are identified and opened widely. An attempt is made to remove the talus as one piece. For all cases in this cohort, the talus was removed completely. Once the talus is removed, the calcaneus is positioned posteriorly in the ankle mortise and held in position with a threaded K-wire from the heel through the calcaneus and into the distal tibia. In the case of residual forefoot deformities, additional procedures, typically lateral column osteotomies, can be performed. A short leg cast is then applied for a total of 6 weeks, with one cast change performed at 3 weeks to remove the K-wire. Postoperatively, an ankle foot orthosis (AFO) is used during the day and night to maintain correction.

Statistical analysis was performed with the SPSS software package (version 25.0) and consisted of descriptive statistics, chi-square with continuity correction for categorical variables, Spearman correlation and Mann-Whitney for continuous variables (Shapiro-Wilk normality test was done before to verify if a parametric test would be applied), with P ​< ​.05 reaching significance.

Results

This study included 31 feet (19 patients), comprising 18 feet with MM and 13 feet with ART. The gender ratio (10 male/9 female) and affected side (16 left and 15 right feet) were similar. In this cohort, mean age at talectomy was 4.16 years (range 0.77–9.99, SD 2.46) and the mean follow-up since talectomy surgery was 15.71 years (range 6.32–36.51, SD 8.43). Table 1 lists the diagnosis, laterality, prior surgeries, age at talectomy, concomitant procedures, deformity recurrence, further surgeries, deformity at last visit, final grade, FMS, and years of follow-up. Talectomy was performed as a salvage procedure in 80.6% of the cases (25 feet). A postero-medial release (PMR) or a posterior medial lateral release (PMLR) was the initial surgery in 15 (48.4%) patients. A third procedure was not required in 16 feet (51.6%) and 1 foot (3.2%) had a fourth surgery. Six feet (19.4%) had a primary talectomy and 50% (3/6) of them did not require a second surgery. The mean number of surgeries per foot was 2.4 (1–4, SD 0.7).

Table 1.

Patient demographics, diagnosis and functionality.

Patient	Diagnosis	Side	Surgeries before talectomy	Age at talectomy	Concomitant procedures	Deformity recurrence	Further surgery	Deformity at last visit	Final result	FMS	Follow-up duration
1	ART	L	(1) TA rel, (2) PMLR	4.46	Evans Ost ​+ ​AT rel	Add-sup		Moderate add-sup	Poor	1.1.1	6.88
		R	(1) TA rel, (2) PMLR		Evans Ost ​+ ​AT rel	Add-sup		Severe add-sup ​+ ​DB	Poor
2	MM	L		1.08					Good	1.1.1	20.88
3	MM	L	PMR	4.21					Good	6.6.6	36.51
		R	PMR			Valgus	Distal tibia varus ost		Good
4	MM	R	PMLR	3.62	Lichtblau				Good	1.1.1	20.65
5	MM	L		1.31		Equinovarus		Moderate equinovarus	Poor	1.1.1	21.30
6	MM	L	(1) TA rel ​+ ​AT rel, (2) PMLR	5.74	CCW				Good	1.1.1	9.40
		R	(1) TA rel ​+ ​AT rel, (2) PMLR		CCW	Cub exostosis	CS ​+ ​bursa exc		Good
7	ART	L	(1) TA rel, (2) PMLR	6.24	2° extensor tenotomy				Good	6.6.6	8.71
		R	(1) TA rel, (2) PMLR		2° extensor tenotomy				Good
8	ART	L	PMLR	2.55		Equinus	Distal tibia extension ost	Moderate equinus	Poor	2.1.1	8.71
		R	PMLR			Equinus	Distal tibia extension ost	Moderate equinus	Poor
9	ART	L	PMR	6.56	CCW ​+ ​FHL rel	Add-sup		Minor add-sup	Good	6.6.6	10.64
		R	PMR		CCW ​+ ​FHL rel				Good
10	MM	R		1.12		Add-sup		Moderate add-sup	Poor	1.1.1	21.15
11	ART	L	PMLR	1.70	Plantar rel				Good	3.1.1	25.33
12	MM	R		0.77	Lichtblau	Equinus	Posterior rel	Severe equinus	Poor	1.1.1	19.17
13	MM	L	PMLR	6.23		Add-sup		Minor add-sup	Good	1.1.1	14.10
		R	PMLR			Add-sup		Minor add-sup	Good
14	MM	L	(1) TA rel, (2) PMLR	2.25	Lichtblau				Good	3.3.1	15.92
		R	(1) TA rel, (2) Posterior rel		Lichtblau ​+ ​TA rel				Good
15	MM	L	PMLR	2.97		Add-sup		Minor add-sup	Good	1.1.1	21.27
		R	PMLR						Good
16	MM	L	(1)PMLR, (2) PMLR rev ​+ ​Lichtblau	4.17	Navicular exc ​+ ​CCW				Good	5.5.1	16.45
17	MM	R		1.14		Add-sup	(2)CCW, (3)CS ​+ ​plantar rel			2.2.1	7.27
		L				Add-sup	CCW
18	ART	R	PMLR	9.99						6.6.6	25.28
		L	PMLR
19	ART	R	(1) TA rel, (2) PMLR	5.33	CCW	Add-sup		Minor add-sup		6.6.6	6.32
		L	(1) TA rel, (2) PMLR		CCW

MM, myelomeningocele; ART, arthrogryposis; L, Left, R, Right; (1): first surgery, (2): second surgery, (3): third surgery, PMLR, postero-medial-lateral release, PMR, postero-medial release, TA, tendon Achilles, AT, anterior tibialis, Rel, release, Rev, revision, CCW, cuboid closing wedge osteotomy, Ost, osteotomy, FHL, flexor hallucis longus, Exc, excision, Cub, cuboid, CS, cuboid shaving, Add-sup, adductus-supination, DB, dorsal bunion.

Isolated talectomy was performed in 15 feet (48.4%), and the remaining 16 feet (51.6%) had concomitant procedures. Osteotomies were combined with talectomy in 13 feet (41.9%), with the most common being a cuboid closing wedge osteotomy (7/13, 53.8%). Concomitant lateral column shortening was required to achieve correction in 81.8% (9/11) of feet that had talectomy as a third surgery while 80% (16/20) of feet that had talectomy as first or second surgery did not have a combined osteotomy. The odds ratio (OR) of needing concomitant lateral column shortening procedure was 6.23 (1.60–24.17) when talectomy was performed as a third surgery (P ​= ​.001).

Superficial infection occurred in 3 feet (9.7%), and no other early complications were reported. Good and stable correction was obtained in 14 feet (45.2%). Deformity after talectomy, classified as recurrence, was identified in 17 feet (54.8%). The most common residual deformities were adduction and/or supination (11 feet, 35.5%), followed by equinus (3 feet, 9.7%). Further surgery for deformity correction, such as distal tibia varus osteotomy, cuboid shaving with bursa excision, distal tibia extension osteotomy, posterior release, cuboid closing wedge osteotomy, and plantar release was required in 7 feet (22.6%), and the mean interval between talectomy and the next procedure was 2.87 years (range 1.38–4.50).

At the last clinical visit, a good result (plantigrade or with only a minor deformity) was found in 24 feet (77.4%). Moderate or severe deformity was present in 7 feet (22.6%) and graded as a poor result. Sporadic ankle pain was mentioned by 2 patients (3 feet, 9.7%); difficulty fitting in regular shoes was reported in 2 feet (6.5%); 4 feet (12.9%) were classified as unbraceable at final follow-up.

Two patients (3 feet, 9.7%) presented with skin breakdown during follow-up. In patient 4, where the foot was plantigrade and braceable, a chronic ulcer developed following an acute trauma requiring treatment. The other 2 feet (patient 6), both defined as plantigrade and braceable at the last visit, presented with bilateral posterior hindfoot pressure sores related to decubitus pressure after prolonged hospitalization in a psychiatric facility. Since all reported sores were not related to foot deformities, these 3 feet were not classified as poor result.

Among the patients that had talectomy performed before five years old (19 feet), poor results at final follow-up (average of 18.6 years) were found in 7 (36.8%) feet. All patients (12 feet) that had surgery after five years old showed a good result at final follow-up (average 12.4). An older age at talectomy surgery was associated with good results at the last clinical visit (P ​= ​.03; good result: mean age 4.66 years, SD 2.49; poor result: mean age 2.46 years, SD 1.52). There was no significant difference between length of follow-up for patients treated with talectomy before and after age five (P ​= ​.12). Additionally, a primary talectomy was associated with more subsequent surgeries per foot when compared with salvage talectomies (P ​= ​.03; primary: 0.7 surgeries, SD 0.8; salvage: 0.2 surgeries, SD 0.4), while primary talectomy was associated with fewer total surgeries per foot than salvage talectomy (P ​= ​.003; primary: 1.7 total surgeries, SD 0.8; salvage: 2.6 total surgeries, SD 0.6).

Patients with baseline FMS of 3-1-1 or higher had satisfactory and stable corrections after talectomy in 71.4% (10/14) of the cases while a recurrence rate of 76.5% (13/17) was observed in patients with FMS lower than 3-1-1 (P ​= ​.008, OR 2.67, CI 1.12–6.38). Additionally, a FMS of 3-1-1 or higher was associated with a good outcome (P ​= ​.022, OR 2.40, confidence interval (CI): 1.49–3.85). All poor results (7 feet, 22.6%) were found in patients with baseline FMS lower than 3-1-1.

When comparing the results according to the diagnosis (MM or ART), the only significant differences were the mean age at talectomy (MM 3.17 ​yrs; ART 5.53 ​yrs; P ​= ​.006) and the walking ability (FMS 3-1-1 or higher: MM 27.8%, 5/18; ART 69.2%, 9/13; P ​= ​.022). The mean number of surgeries per foot was similar (MM 2.28, ART 2.62; P ​= ​.18). Primary diagnosis did not influence deformity recurrence (MM 55.6%, 10/18; ART 53.8%, 7/13; P ​= ​.92), need for further surgery (MM 27.8%, 5/18; ART 15.4%, 2/13; P ​= ​.41) or outcome at last visit (good result: MM 83.3%, 15/18; ART 69.2%, 9/13; P ​= ​.35).

There was no significant difference in length of follow-up for good versus poor results (P ​= ​.39; good: mean 16.42 ​yrs, SD 8.83; poor: mean 13.25 ​yrs, SD 6.88). Additionally, a longer follow-up was correlated with fewer surgeries per foot (Spearman r ​= ​−0.59, P ​< ​.001).

Discussion

The ultimate goal for treatment of clubfoot in patients with MM or ART is a stable, braceable, and plantigrade foot [8,13,14]. To safely achieve correction in these resistant clubfoot, more aggressive procedures such as talectomy may be required while triple arthrodesis and gradual correction with external fixators should be reserved for older patients [9,10]. Traditionally, less radical surgery is performed before talus excision. However, if a child presents with severe deformity and fails all conservative measures, talectomy may be chosen as a primary procedure, though this only occurred in 6 feet (19.4%) in this study [10,15]. Figure 1, Figure 2 represent a possible course of treatment for patients with ART or MM, respectively.

Figure 1.

A) TEV in a newborn with arthrogryposis. Initial treatments: casting and Achilles tenotomy followed by posterior medial lateral release with bilateral talectomy at 5.3 years old. B) and C) Final result at 11.6 years old with FMS of 6-6-6. TEV, talipes equinovarus; FMS, Functional Mobility Scale.

Figure 2.

A) TEV in a newborn with spina bifida. Initial treatments: casting and Achilles tenotomy followed by right posterior release and left posterior medial lateral release with a bilateral talectomy at 2 years old. B) and C) Result at 18 years old, plantigrade and braceable with no pressure sore and FMS of 3-3-1. TEV, talipes equinovarus; FMS, Functional Mobility Scale.

Previous literature suggests primary talectomy was noted to have better results than salvage talectomies in arthrogryposis patients, with decreased recurrence rates, less complications and reduced number of surgeries when done as a primary procedure [3,9]. This contradicts our finding that the number of subsequent surgeries per foot was increased in primary versus salvage talectomies (P ​= ​.03).

Despite the promising short-term results, both hindfoot equinovarus and combined adductus and supination were observed in the longer term [3,14,16]. To prevent deformity recurrence, it is suggested to perform the surgery closer to when the patient normally begins to stand. Orthoses for daytime walking and resting night splints should be used throughout the growth period to avoid recurrence [17]. Green et al. reported that relapse into equinovarus or cavus occurred between two and six years later, similar to our findings [15]. In our study, further surgery (listed in Table 1) for deformity correction was performed between 1.38 and 4.50 years after talectomy (mean 2.87 years).

Combined forefoot adduction and supination is the most common residual deformity reported as recurrence, most often due to a fibrotic and contracted tibialis anterior [3,14,15]. Legaspi, in a 20-year follow-up study of 24 feet, reported recurrence of hindfoot deformities in 16 feet (67%). Additional surgery to correct forefoot deformity was not considered failure or poor outcome, since talectomy was carried out to correct the hindfoot only [4]. This idea has previously been supported by Dias and Stern [6]. Talectomy is effective for correction of the hindfoot varus and equinus, and surgeons need to understand that the associated forefoot problems should be treated as separate entities to achieve complete correction [3,6,18].

Many concomitant procedures have been reported as options with talectomy to achieve full correction [3,18]. The navicular can be excised if necessary to obtain adequate posterior positioning of the calcaneus [18]. Lateral column shortening helps correct residual adductus and can be done by cuboid decancellation, cuboid closing wedge, excision of the anterior calcaneus (Lichtblau procedure), or fusion of the calcaneocuboid joint (Evans procedure) [3,18]. Pirpiris compared the results of 14 isolated talectomies and 17 talectomies combined with calcaneocuboid fusions, and medium-term results suggested the concomitant procedure was associated with decreased need for revision, postoperative equinus, varus, adductus, and supination deformities [16]. Among the feet with deformity recurrence in our study, 58.8% (10/17) had an isolated talectomy. On the other hand, 64.3% (9/14) of feet that showed satisfactory and stable correction after talectomy had a concomitant procedure. However, the presence of concomitant surgery did not correlate with deformity recurrence after talectomy (P ​= ​.2).

Two patients (3 feet, 9.7%) mentioned sporadic ankle pain during long-term follow-up visits. Neither of them had pain on visit dates or required further treatment and all 3 feet were braceable and plantigrade. One (patient 18) stood all day in his job and reported full relief after reducing hours of work. The other (patient 16) was considered obese and described mild pain only with prolonged standing or intensive activities. For those reasons, both patients were still classified as having good result despite the reported pain.

The low incidence of pain after talectomy may be related to the limited ambulatory demands of these patients, who usually present with associated disabilities [18]. In patients with MM, neurosegmental level is the primary determinant of walking ability and physical function. However, many of them have foot deformities that can often limit function, preclude an effective gait, and lead to pressure sores [1]. Even for nonambulatory patients, foot appearance, shoe wear, and bracing can become important issues. The early use of standers and gait trainers in childhood should be encouraged even in more compromised patients. Our results suggest that walking ability has a significant role in preventing recurrence. A FMS of 3-1-1 or higher had a positive correlation with stable corrections after talectomy (P ​= ​.008) and also with a good outcome at the last visit (P ​= ​.022).

In this study, all poor results were found in patients that had talectomy before five years old (7/19, 36.8%), while 100% of patients older than five at talectomy showed good results at final follow-up (P ​= ​.026). However, this may partially be due to patients with more severe deformities presenting for treatment at a younger age. This contrasts with previous recommendations that talectomy is most likely to give good results when performed between the ages of one and five years old [10,17], while other studies did not find significant association between age at the time of surgery and outcome [9,13].

Some limitations of this study should be discussed. First, 22 feet, or 42% of the potential cohort, were excluded for insufficient follow-up, and it is unclear how this would have impacted the results. Another limitation of this study is the lack of radiological evaluation, which was not included because it was not routinely performed during clinical visits and does not adequately correlate with clinical outcomes. In many cases, concomitant procedures were performed, limiting the generalizability of talectomy results. Additionally, the retrospective nature of this study prohibits the distinction of early relapse from correction recurrence in the assessment and classification of residual deformity. Finally, rare use of talectomy in practice (5.6% of the initial population of 944 clubfoot with MM or ART) limits the sample size but demonstrates that talectomy is a reserved treatment for clubfoot and surgeons should first consider a less aggressive approach in almost all cases.

In conclusion, our long-term results reinforce talectomy as an important option for treatment of severe TEV. Good results were observed in longer follow-up patients and a satisfactory outcome remains until adulthood, with the best results observed in patients with a FMS greater than 3-1-1. Further surgery was not required in 77.4% of the cases, and 87.1% of the feet were braceable. Despite a high recurrence rate that should be expected for these resistant deformities, good outcomes can be achieved for the majority of patients who undergo talectomy.

Additional links

• •POSNAcademy: A Novel Technique to Remove Plaster Ponseti Clubfoot Casts.
• •AAOS Orthopaedic Video Theatre: Posteromedial Release for Correction of Neglected Rigid Clubfeet.

Consent for publication

Complete written informed consent was obtained from the patient, their guardian or legal representative for the publication of this study and accompanying images.

Author contributions

Pedro Poggiali: Writing – original draft, Methodology, Investigation, Formal analysis, Data curation, Conceptualization. Jared M. May: Writing – review & editing, Writing – original draft, Investigation, Formal analysis, Data curation. Jill E. Larson: Writing – review & editing, Validation, Supervision, Project administration, Methodology, Investigation, Data curation, Conceptualization. Luciano Dias: Validation, Supervision, Methodology, Investigation, Formal analysis, Conceptualization.

Funding

No intra- or extramural funding was used to support this research.

Declaration of competing interests

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.