6 Original article

Does the Ponseti technique affect the vascular development in patients with congenital talipes equinovarus? Timur Yildirima, Aysegul Bursalib, Murat Tonbulc, Secil S. Sakizlioglua and Yalkin Camurcua In this study, we have evaluated the changes observed in the main arteries of the foot before and after the Ponseti technique. Arterial structures of seven patients were examined using Doppler ultrasound and the parameters studied included the course of the arteries, lumen filling, flow direction, pattern, and velocity. Before the treatment, the side with congenital talipes equinovarus deformity showed decreased blood flow in all arteries, except for the posterior tibial artery. At the second examination, the increase in the flow velocity of both arteries except tibialis anterior arteries was statistically significant. The Ponseti method results in normalization of the arterial structures

Introduction Congenital talipes equinovarus (CTEV) is the most frequent congenital anomaly of the musculoskeletal system, with a worldwide incidence of one per 1000 live births, with some geographical variations [1]. CTEV can occur isolated or in conjunction with other conditions such as arthrogryposis multiplex congenita, myelodysplasia, fibular hemimelia, etc. [2,3]. Several hypotheses have been presented to explain the etiology of CTEV including developmental arrest of the neuromuscular unit during intrauterine development, compression effect in the intrauterine cavity because of inadequate space for the fetus, or genetic mutations, although none of them have been confirmed as yet [4–6]. The Ponseti technique, involving manipulation and casting, has a widespread acceptance with increasing rates of use for the treatment of CTEV [1,7]. This technique allows satisfactory correction of the anatomical deformation of the foot and reduces the need for surgery. Observation of abnormally small or absent dorsalis pedis artery and/or posterior tibial artery (rarely) during CTEV correction surgery and related complications has led to researches examining the association between the etiology of CTEV and vascular developmental defects [3,8–10]. Ultrasonographic (US) examination of healthy feet indicated the absence of the anterior tibial artery for 2.2% [11], whereas higher numbers were reported for CTEV [9,12–14]. The longer the deformity remains, that is, the older the patient is, the more common the vascular defects [11,13]. CTEV cases with arterial anomalies have a tendency for recurrence [14]. In addition, higher 1060-152X © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins

in extremities with congenital talipes equinovarus. J Pediatr Orthop B 24:6–10 © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins. Journal of Pediatric Orthopaedics B 2015, 24:6–10 Keywords: Ponseti method, talipes equinovarus, ultrasound evaluation, vascular development a Baltalimani Bone and Joint Diseases Education and Research Hospital, bPrivate Practice, Istanbul and cReyap Hospital, Corlu-Tekirdag, Turkey

Correspondence to Timur Yildirim, MD, Baltalimani Bone and Joint Diseases Education and Research Hospital, Rumeli Hisari Cad. No: 62, 34470 Sariyer-Istanbul, Turkey Tel: + 90 212 323 70 75; fax: + 90 212 323 70 82; e-mail: [email protected]

incidences of vascular abnormalities were detected in the parents of children with CTEV when compared with controls [15]. Although arteriography is the gold-standard technique for examination of the arteries of the lower extremity, its invasive nature limits routine use in pediatric populations [9,12,13,16,17]. Alternative imaging modalities include continuous-wave Doppler, color triplex flow Doppler, and threedimensional magnetic resonance (MR) angiography without contrast enhancement [3,8,10,11,15,16,18,19]. Previous works suggest that continuous-wave Doppler examination may yield misleading results [10,16]. Although three-dimensional MR angiography without contrast is a noninvasive imaging technique, its use in small children usually requires general anesthesia [14]. Ideally, a good knowledge of the anatomy of the vasculature of the lower extremity may provide valuable prognostic information for the orthopedic surgeon before manipulation and casting, especially in terms of determining the speed of correction (some cases may require a more gradual correction) or risk of recurrence. This information is also important before surgery. Although vascular abnormalities were reported to increase with age, literature data on preintervention (i.e. before manipulation and casting) examination of the vasculature of the lower extremity and the effect of treatment modalities on vascular development are rather scarce. DOI: 10.1097/BPB.0000000000000105

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Vascularity after Ponseti technique Yildirim et al. 7

This relative lack of data is particularly relevant for the Ponseti technique, where no specific literature information exists on the development of the arteries supplying the feet during the reshaping process of the feet and legs with manipulation and gradual correction. To our knowledge, no other studies have previously used the color triplex Doppler technique for the comparison of anterior tibial, posterior tibial, and dorsalis pedis arteries before and after correction of CTEV deformity with the Ponseti technique. In this study, our aim was to assess the changes and compare the anatomical and functional characteristics of the anterior tibial, posterior tibial, and dorsalis pedis arteries using the color triplex flow Doppler method before and at least 6 months after correction in patients with unilateral CTEV.

Materials and methods A total of seven treatment-naive children with unilateral CTEV and no other medical conditions were included in this prospective study between September 2010 and March 2012. The study protocol was approved by the local ethics committee and informed consent was obtained from all parents. The CTEV deformity was at the right side in four children and at the left side in three children. All patients were male, with an average age of 66.7 days (range, 14–186 days) at the time of initial assessment (Table 1). On average, the second assessment was performed 12 months after the completion of casting (range, 7.5–24 months). Before manipulation, all feet were categorized using the Pirani classification. The average Pirani score was 5.4 (range, 3–6). Manipulation and casting were performed weekly in accordance with the Ponseti technique. For percutaneous achillotomy, local anesthesia was administered in the outpatient unit. The final cast remained in place for 3 weeks. Following this, a Denis Browne bar and special boots were used for 23 h and the duration of these sessions was reduced every 3 months. At the time of the second Doppler examination, all patients were using the Denis Browne bar and strait-last boots. No recurrence occurred. Subsequently, two cases (case 2 and 5) that had stopped using the orthosis had recurrent Table 1

Family history of CTEV was negative in all patients. All patients were evaluated by the same radiologist (S.S. S.) using a Toshiba Aplio XG (Toshiba Medical Systems Corporation, Tochigi, Japan) device. Bilateral color Doppler US was performed to examine the arterial structures of the crural region including the anterior tibial artery in the proximal third of the anterior leg, dorsalis pedis artery in the intermetatarsal region at the distal part of the first and second metatarsal bones of the dorsum of the foot, and the segment of the posterior tibial artery running posterior to the medial malleolus. Fractional measurements were performed as the babies were calm and sleeping so as not to lead to false alterations in the blood flow rates. Parameters examined included the course and diameter of the arteries, lumen filling (in color mode), flow direction, and flow pattern and velocity (spectral analysis). Extremity with CTEV was compared with the unaffected side. All data were analyzed using the statistical software SPSS 15.0 (SPSS Inc., Chicago, Illinois, USA). For the statistical comparisons, nonparametric Mann–Whitney U-test and Wilcoxon tests were used. Statistical significance was defined as P value less than or equal to 0.05.

Results None of the seven babies with unilateral CTEV deformity had absence of anterior tibial, posterior tibial, or dorsalis pedis artery. However, the side with the CTEV deformity showed decreased blood flow in all arteries, except for the posterior tibial artery, but this difference was not statistically significant (P > 0.05). The average blood flow velocity in the anterior tibial, posterior tibial, and dorsalis pedis arteries was 31.85 cm/s (13–51), 36.71 cm/s (27–48), and 15.57 cm/s (4–24), respectively. The corresponding figures on the side without deformity were 32.71 cm/s (13–58), 35 cm/s (22–46), and 16.42 cm/s (5–25), respectively. Therefore, percent velocities of the arteries in the CTEV side as compared with the unaffected side were 97.4, 104.9, and 94.8%, respectively (P = 0.9, 0.65, and 0.65).

Characteristics of the patients

Case number 1 2 3 4 5 6 7

deformity that was corrected with casting without the need for surgery.

Sex

Side

Pirani score

Age (days)

Number of casting

Duration of casting (weeks)

Interval between two ultrasound evaluations (months)

M M M M M M M

R R R L L L R

5 6 6 6 4 6 5

186 14 29 19 142 39 38

6 3 4 4 5 3 4

9 6 7 7 8 6 7

7.5 7.5 13 12 12 24 10

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8 Journal of Pediatric Orthopaedics B 2015, Vol 24 No 1

At the second US examination, the increase in flow velocity of both arteries, except tibialis anterior arteries, was statistically significant (P < 0.05) Following casting with the Ponseti technique, secondary US measurements showed flow velocities of 34.86 cm/s (13–55), 72.71 cm/s (57–100), and 34 cm/s (6–70) for the same arteries in the side with the deformity, respectively. After casting, the corresponding figures for the side without deformity were 37.86 cm/s (14–60), 74.29 cm/s (48–110), and 32.86 cm/s (8–66), respectively. The percent velocities in the side with the deformity compared with the side without deformity were 92, 97.9, and 103.5%, respectively (P = 0.56, 0.85, and 0.75). A statistically nonsignificant increase in the flow velocity in the dorsalis pedis artery was noted. No arteries showed retrograde flow. The overall results are shown in Table 2.

Discussion Talipes equinovarus deformity represents the most common congenital anomaly of the musculoskeletal system and the Ponseti method aimed at the gradual correction of this deformity has revolutionized the management of this condition worldwide, providing many children with the opportunity to walk with plantigrade feet [7,20–23]. However, surgery has not been able to reproduce the same satisfactory success rates in children with talipes equinovarus deformity [1]. Inadequate circulation in the arteries supplying feet may increase the complication rate and adversely affect the outcome of surgery [3,8,10,11,14,15]. Absence or hypoplasia of the dorsalis pedis artery and absence of the posterior tibial artery have been reported to occur in feet with CTEV [9, 11,16,17,24,25]. Absence of the dorsalis pedis artery has been reported in 6.7–86% of the patients with CTEV [11–13]. Severity of the deformity or presence of concomitant anomalies generally correlates with the absence of arteries or inadequate circulation [3,11,14,18]. Hypoplasia of the anterior tibial artery is more common than the posterior tibial artery [3,10,14]. In the absence of the anterior tibial artery, quick dorsiflexion with manipulation may place tension stress on the single artery and result in the disruption of the circulation. Particularly during the posteromedial relaxation, injury to the posterior tibial artery may lead to serious complications including extremity loss. In this prospective study, before Table 2

casting, vascularization of the extremity was evaluated to determine existing vascular or flow abnormalities and to monitor the vascular development of the extremity following corrective manipulation. To our knowledge, no previous studies have examined the vascular development of the extremity in patients with CTEV undergoing Ponseti correction. Although arteriography represents the gold-standard method for examination of the arterial structures, its use in small children poses significant challenges [10]. However, three-dimensional MR angiography without contrast enhancement requires general anesthesia [17]. Therefore, color Doppler examination was performed to assess the vascular development in our patients. In support of this, color flow Doppler examination has also been reported to be similarly effective in estimating the prevalence of arterial defects as arteriography, despite the arteriography’s role as the goldstandard method for the evaluation of arterial abnormalities and flow direction [10]. In most of the angiography studies, deficient arteries are defined as those that are absent or abnormally small or have retrograde flow. Because the abnormally small arteries detected by angiography may not be detected by pulsed color flow Doppler US, the reported prevalence of the arterial deficiencies should be similar to these two techniques. In our study of unilateral CTEV cases, the unaffected extremity was used as the control. Although several authors have maintained that the unaffected extremity in cases with unilateral CTEV cannot be used as the control on the basis of the difference observed between the unaffected foot of unilateral CTEV cases and the feet of healthy children in terms of the results of pedographic and gait analyses, others have used the unaffected foot as the control considering the need for a comparison [14,26–28]. Katz et al. [10] have found no difference in the results of arterial examination with pulsed color Doppler and flow Doppler between the contralateral foot of unilateral CTEV patients and the feet of healthy children, thus concluding that the contralateral foot can be used as the control. In our study, the color flow Doppler before casting showed a lower flow velocity in the anterior tibial and dorsalis pedis arteries of the affected side as compared with controls, whereas the flow velocity in the posterior tibial artery was higher in the affected side,

Mean blood flow velocity at first and second evaluations The average flow rate in the first review (cm/s)

Arteria tibialis anterior Arteria tibialis posterior Arteria dorsalis pedis

a

The average flow rate in the last review (cm/s)

CTEV side

Nonaffected side

%

P

CTEV side

Nonaffected side

%a

P

31.85 36.71 15.57

32.71 35.00 16.42

97.4 104.9 95.0

0.90 0.65 0.65

34.86 72.71 34.00

37.86 74.29 32.86

92.0 97.9 103.5

0.56 0.85 0.75

CTEV, congenital talipes equinovarus. a CTEV: nonaffected side blood flow ratio.

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Vascularity after Ponseti technique Yildirim et al. 9

which might represent a compensatory mechanism for the decreased blood flow in the dorsalis pedis artery.

the determination of the blood flow velocity and direction. Therefore, only the latter two were examined.

In this study, the results of color Doppler examination of the arteries following the completion of the correction with the Ponseti method showed a gradual improvement not only in the bony and articular structures but also in the development of the arteries. The flow velocity of the dorsalis pedis artery showed a 104% improvement following the treatment compared with the unaffected side. The improvement compared with the baseline was 9%. The increase in flow velocity of the dorsalis pedis artery in the side with CTEV was 118.36% when compared with the baseline (before treatment). The corresponding figures for the anterior tibial and posterior tibial arteries were 9.42 and 98%, respectively. A similar development was also found in the unaffected side as follows: 100, 15.74, and 112.25% in the dorsalis pedis, anterior tibialis, and posterior tibialis arteries, respectively.

Limitations of our study include the small sample size as well as the absence of controls with healthy feet. Because of technical difficulties associated with study measurements in very small children, contralateral feet were used as the controls.

In a previous study, although no difference was found between the treated and the unaffected side in terms of the results of arterial examination [19], it should be noted that a technique other than the Ponseti method was used for correction in their patients in conjunction with an US technique with questionable efficacy, that is, continuous flow US method, for arterial assessments. In addition, some patients underwent physiotherapy. In another study with flow Doppler US, the anterior tibial artery was absent in a single case, and although a decrease in the flow velocity in proportion to the severity of the deformity was observed, no significant differences were observed between the affected and the unaffected sides in terms of flow velocity. In that study, a wide range of patients from different age groups were included, most of whom had already undergone surgery, and were treated using the Kite technique for casting [29]. No significant changes in the anterior tibial artery were found, in contrast with significant changes in the other arteries examined. The presence of a similar developmental course in the unaffected side suggests that this change might represent the natural development. Although the small sample size of this study is a statistical disadvantage, the percent increase in the flow velocity of the dorsalis pedis artery is noteworthy and suggests the emergence of a developmental activity of the dorsalis pedis artery targeting normal development in parallel with the correction in the foot deformity. Our literature search has yielded only a single study involving coexamination of the anterior tibial and dorsalis pedis arteries in these cases where the hypoplastic anterior tibial artery was reported [13]. Participants were older and had longer duration of deformity, limiting the comparability to our findings. Similarly, no other studies comparing pretreatment and post-treatment (i.e. before casting and after the completion of the treatment) assessments were found. In small children, vessel diameter measurement is technically challenging and does not yield as much information as

Despite these limitations, our study is the first of its kind in the sense that it involves two sets of measurements to assess the arterial development by comparing pretreatment (i.e. before manipulation and casting) and posttreatment results. The Ponseti method not only allows for the reshaping of the bony and articular structures but also facilitates arterial development. Our results suggest that hypoplasia or absence of the dorsalis pedis artery reported previously in up to 82% of the cases may actually represent a process co-occurring along with the deformity, where vascular development may be hindered. In this respect, the reliability of the technique used for arterial assessments is of high relevance as some of the previous work on this topic involves the use of techniques such as continuous flow Doppler with questionable efficacy. Conclusion

The Ponseti method results in normalization of the arterial structures in extremities with CTEV. Further studies are warranted to confirm our findings.

Acknowledgements Conflicts of interest

There are no conflicts of interest.

References 1

Dobbs MB, Gurnett CA. Update on clubfoot: etiology and treatment. Clin Orthop Relat Res 2009; 467:1146–1153. 2 Wynne-Davies R. Family studies and the cause of congenital club foot. talipes equinovarus, talipes calcaneo-valgus and metatarsus varus. J Bone Joint Surg Br 1964; 46:445–463. 3 David RH, Packard DS Jr, Levinsohn EM, Berkowitz SA, Aronsson DD, Crider RJ Jr. Ischemic necrosis following clubfoot surgery: the purple hallux sign. J Pediatr Orthop B 2004; 13:315–322. 4 Gordon N. Arthrogryposis multiplex congenita. Brain Dev 1998; 20:507–511. 5 Dunn PM. Congenital postural deformities: perinatal associations. Proc R Soc Med 1972; 65:735–738. 6 Gurnett CA, Alaee F, Kruse LM, Desruisseau DM, Hecht JT, Wise CA, et al. Asymmetric lower-limb malformations in individuals with homeobox PITX1 gene mutation. Am J Hum Genet 2008; 83:616–622. 7 Morcuende JA. Congenital idiopathic clubfoot: prevention of late deformity and disability by conservative treatment with the Ponseti technique. Pediatr Ann 2006; 35:128–130, 132–136. 8 Hootnick DR, Packard DS Jr, Levinsohn EM, Crider RJ Jr. Confirmation of arterial deficiencies in a limb with necrosis following clubfoot surgery. J Pediatr Orthop B 1999; 8:187–193. 9 Hootnick DR, Levinsohn EM, Crider RJ, Packard DS Jr. Congenital arterial malformations associated with clubfoot: a report of two cases. Clin Orthop Relat Res 1982; 167:160–163. 10 Katz DA, Albanese EL, Levinsohn EM, Hootnick DR, Packard DS Jr, Grant WD, et al. Pulsed color-flow Doppler analysis of arterial deficiency in idiopathic clubfoot. J Pediatr Orthop 2003; 23:84–87.

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10 Journal of Pediatric Orthopaedics B 2015, Vol 24 No 1

11 12 13 14

15

16

17

18

19

Edelson JG, Husseini N. The pulseless club foot. J Bone Joint Surg Br 1984; 66:700–702. Ben-Menachem Y, Butler JE. Arteriography of the foot in congenital deformities. J Bone Joint Surg Am 1974; 56:1625–1630. Greider TD, Siff SJ, Gerson P, Donovan MM. Arteriography in club foot. J Bone Joint Surg Am 1982; 64:837–840. Merrill LJ, Gurnett CA, Siegel M, Sonavane S, Dobbs MB. Vascular abnormalities correlate with decreased soft tissue volumes in idiopathic clubfoot. Clin Orthop Relat Res 2011; 469:1442–1449. Muir L, Laliotis N, Kutty S, Klenerman L. Absence of the dorsalis pedis pulse in the parents of children with club foot. J Bone Joint Surg Br 1995; 77:114–116. Sodre H, Bruschini S, Mestriner LA, Miranda F Jr, Levinsohn EM, Packard DS Jr, et al. Arterial abnormalities in talipes equinovarus as assessed by angiography and the Doppler technique. J Pediatr Orthop 1990; 10:101–104. Dobbs MB, Gordon JE, Schoenecker PL. Absent posterior tibial artery associated with idiopathic clubfoot. A report of two cases. J Bone Joint Surg Am 2004; 86-A:599–602. Kruse L, Gurnett CA, Hootnick D, Dobbs MB. Magnetic resonance angiography in clubfoot and vertical talus: a feasibility study. Clin Orthop Relat Res 2009; 467:1250–1255. Vispo Seara JL, Arnold JP, Lorenz M, Krauspe R. Arterial blood flow in congenital idiopathic clubfoot [article in German]. Z Orthop Ihre Grenzgeb 1996; 134:533–536.

20 21

22 23 24 25

26

27 28

29

Ballantyne JA, Macnicol MF. Congenital talipes equinovarus (clubfoot): an overview of the aetiology and treatment. Curr Orthop 2002; 16:85–95. Morcuende JA, Dolan LA, Dietz FR, Ponseti IV. Radical reduction in the rate of extensive corrective surgery for clubfoot using the Ponseti method. Pediatrics 2004; 113:376–380. Dobbs MB. Clubfoot: etiology and treatment: editorial comment. Clin Orthop Relat Res 2009; 467:1119–1120. Garg S, Dobbs MB. Use of the Ponseti method for recurrent clubfoot following posteromedial release. Indian J Orthop 2008; 42:68–72. Bursali A, Yurdoglu C. Absent posterior tibial artery in a case with clubfoot. Acta Orthop Traumatol Turc 1992; 26:197–198. Schwering L, Ruppert R, Reichelt A. Aplasia of the posterior tibial artery in a child with idiopathic clubfoot [article in German]. Orthopade 2003; 32:437–438. Favre P, Exner GU, Drerup B, Schmid D, Wetz HH, Jacob HA. The contralateral foot in children with unilateral clubfoot: a study of pressures and forces involved in gait. J Pediatr Orthop 2007; 27:54–59. Ippolito E, Ponseti IV. Congenital club foot in the human fetus. A histological study. J Bone Joint Surg Am 1980; 62:8–22. Ippolito E, De Maio F, Mancini F, Bellini D, Orefice A. Leg muscle atrophy in idiopathic congenital clubfoot: is it primitive or acquired? J Child Orthop 2009; 3:171–178. Pinto JA, Blumetti FC, Iha LAN, Terasaka MK, Sodre H, Ishida A, et al. Evaluation of arterial anatomy in congenital clubfoot with color doppler ultrasound. Acta Orthop Bras 2008; 16:270–274.

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Does the Ponseti technique affect the vascular development in patients with congenital talipes equinovarus?

In this study, we have evaluated the changes observed in the main arteries of the foot before and after the Ponseti technique. Arterial structures of ...
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