Comprehensive Outcome

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Editorial Manager(tm) for Journal of Pediatric Orthopaedics B Manuscript Draft Manuscript Number: Title: Comprehensive Outcome and Functional Evaluation of Clubfoot Treatment Article Type: Original Study Keywords: clubfoot, outcome evaluation, gait analysis Corresponding Author: Ken N. KUO, MD Corresponding Author's Institution: National Health Research Institutes First Author: Adam Graf, MS Order of Authors: Adam Graf, MS;Kuan-Wen Wu, MD;Peter A Smith, MD;Ken N. KUO, MD Manuscript Region of Origin: UNITED STATES Abstract: The treatment outcome has been a focus interest in those who manage clubfoot. Due to lack of common language in the treatment outcome, it is necessary to establish a universal recognized quantitative measurement in order to compare and better understand the treatment outcome. The outcome is not merely a morphological and radiographic, but also should include functional and quality of life measurement. In this article, we like to outline our methods in evaluation of long term outcome including adulthood in this congenial disease. The pre-treatment classifications are important in prognosticating the results. The physical examinations and plain radiographs in standing position are two basic evaluations in clubfoot. Several outcome evaluations were published in the literature. Increasing use of gait analysis is an additional useful technical tool in analyzing the motion of the foot and ankle and its relation to the whole body function. Pedobarography added to the dynamic of the evaluation. Functional quality of life is increasing popularity in measuring the total body functional status and quality of life.

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Comprehensive Outcome and Functional Evaluation of Clubfoot Treatment

Abstract The treatment outcome has been a focus interest in those who manage clubfoot. Due to lack of common language in the treatment outcome, it is necessary to establish a universal recognized quantitative measurement in order to compare and better understand the treatment outcome. The outcome is not merely a morphological and radiographic, but also should include functional and quality of life measurement. In this article, we like to outline our methods in evaluation of long term outcome including adulthood in this congenial disease. The pre-treatment classifications are important in prognosticating the results. The physical examinations and plain radiographs in standing position are two basic evaluations in clubfoot. Several outcome evaluations were published in the literature. Increasing use of gait analysis is an additional useful technical tool in analyzing the motion of the foot and ankle and its relation to the whole body function. Pedobarography added to the dynamic of the evaluation. Functional quality of life is increasing popularity in measuring the total body functional status and quality of life.

INTRODUCTION Talipes equinovarus (clubfoot) is a common, congenital deformity affecting approximately 1-2 in 1000 births. Historically, the treatment of clubfoot deformity was mainly surgical since 40 years ago until Ponseti technique and French method gained wide spread popularity with good results because of improved functional results. The evaluations of the outcome of clubfoot treatment evolved from pure radiographic and morphological to more functional and patient oriented outcome. The extensive soft tissue releases do not always yield satisfactory long-term clinical results. Studies have reported that during adolescent, individuals following treatment might present with residual pain, decrease in strength, and functional deficits as well as gait anomaly. Therefore, the evaluation of children treated for clubfoot must include not only clinical and radiological examination but also functional assessment and quality of life outcome. There were many outcome results published in the literature, however many of them are based on a short term and midterm results. Initially, the outcomes nmostly focused on the morphological change and radiographic improvement. Less attention was paid to the functional results and subjective symptoms. Since the feet are parts of body, the dysfunction of the feet may influence the person’s activity and well being. In this day of patient-centered care, we cannot just look at the foot morphology and radiographic pictures anymore. In addition, with number of treatment outcome studies in the literature, comparing the results of different series is a necessary. The existing quantitative comparisons are not often practical. There should be a universal language in evaluation of ultimate clubfoot treatment outcome including functional and patient perspective.

The purpose of this article is to present a current paradigm for the quantitative comprehensive assessment of clubfoot. A detailed understanding of the outcomes of children and adults with clubfoot is essential to compare treatment methods and identify areas for improvement. In the simplest sense, measurement and descriptions of the foot are meant to determine whether the foot will be useful for a lifetime of good function [1]. The simplest measures and descriptions, particularly in published reports of operative and non-operative treatment, are ratings systems which describe the foot as “plantigrade,” and may even determine the morphology of the hindfoot, midfoot and forefoot separately. Such descriptions are useful to show whether the actual goal of treatment has been successful; that is, the child walks on the bottom of the foot, the foot fits into a shoe, and the general morphology and even motion appear satisfactory. For example, the basic morphology of the foot and minimum amount of motion needed to maintain correction in a brace have been clarified. Subsequently, the average number of relapses experienced during Ponseti management has been decreased. Young children seldom have pain, or difficulty with shoe wear, even when the foot is grossly under or overcorrected. As children grow, morphologic differences in the foot can be measured with a greater degree of accuracy, and begin to have more clinical bearing on the function of the foot. Radiographs, range of motion, and strength measures are more reliable in older children. Even more sophisticated measures of function such as gait analysis and outcomes measures such as the Disease Specific Index (DSI) have been used to differentiate results in children as young as 5 years old.

Since there are numerous reports of overall success of treatment with many different methods, surgical and non-surgical, in children, it has not been until relatively recently, when an emphasis on long term function in adults was introduced, that proponents of Ponseti management have found evidence for improved outcomes in adults with that technique. Ponseti published results in adults using radiographs, physical exam measures, and outcomes measures of satisfaction and work history which reported overall good results. In a study of surgical results in adults with clubfeet Dobbs et al. documented feet with stiffness, pain, and poor positioning.

METHODOLOGY Classification of Infant Clubfeet Classification (or scoring) is helpful in determining a prognosis, plan of treatment and tracking changes in the foot although it is somewhat controversial as to how necessary it is. There are several clubfoot classifications that mainly use a morphologic rating scale rigidity of the foot to describe the prominent features of clubfoot such as cavus, varus, adductus, and equinus [2-5]. The two most common scoring systems are the Pirani and Dimeglio methods. The Pirani Clubfoot Score documents the severity of contracture of three components of the hindfoot and three components of the midfoot[6]. Each is provided with a score of 0 (no abnormality), 0.5 (moderate abnormality), or 1 (severe abnormality). The hindfoot components are: severity of the posterior crease, emptiness of the heel and rigidity of the equinus. The three related to the midfoot are curvature of the lateral border of the foot, severity of the medial crease and the position of the lateral part of the head of the talus. The Dimeglio Clubfoot Score rates the equinus and varus

deviations in the sagittal plane, and supination and adduction of the forefoot in the coronal plane [7, 8]. Of multiple parameters, four are considered of vital importance: 1) equinus deviation in the sagittal plane, 2) varus deviation in the frontal plane, 3) endorotation of the calcaneo-forefoot block in the horizontal plane (supination), 4) adduction of the forefoot in the horizontal plane.

Physical Examination A hands on physical examination performed by a licensed physical therapist or physician is often necessary to provide information about the foot stiffness, range of motion and strength. Anthropometrics such as calf circumference, leg length and foot dimensions can also be useful. The goniometric measures of the foot and ankle that are commonly acquired include: ankle dorsiflexion/plantarflexion, hindfoot inversion/eversion, forefoot adduction/abduction and supination/pronation as well as thigh foot angle and transmaleolar axis. Observations are made of the static weight-bearing alignment, foot morphology and skin integrity (Table 2). Calf atrophy, leg length discrepancy, knee hyperextension and muscle weakness are common characteristics of individuals with clubfeet (Figure 3). Strength can be assessed in a variety of ways such as manual muscle testing (Jones classification) and functional strength assessments (heel raises). Dynomometry can be used for a more consistent, quantitative measure of various types of muscle contractions.

Plain Radiographs

Weight-bearing radiographs are standard of care in the treatment of clubfoot and usually consist of views from the anterior/posterior and lateral directions. Specific measures of importance include quantification of parallelism of the talus and calcaneus in the A/P and lateral planes and the degree of equinus of the calcaneus in relation to the tibia. These can be used to measure bony alignment after treatment during childhood and to detect osteo-arthritis within the joints during adulthood. Following treatment radiographic measurements are usually improved but still abnormal and may correlate with the degree of residual deformity (Figure 1A and B). It is common for there to be residual parallelism of the talus and calcaneus or a reduction of the A/P or lateral talocalcaneal angle. Calcaneal pitch or the angle of the calcaneus with respect to the ground can be abnormally diminished, particularly in surgically treated feet [9] (Table 1). The relation of the first metatarsal to the lateral border of the calcaneus in the A/P plane is a good measure of forefoot abduction or adduction (Figure 2 A and B). The relationship of the first metatarsal to the talar axis on a lateral view (Murray’s Angle) is an indication of residual planus or cavus of the forefoot. The navicular does not ossify typically until 3 years of age, but when the ossified navicular appears it can demonstrate an abnormal dorsal subluxation in relation to the talar head particularly in children treated operatively [10]. Kellgren et al. have developed a useful system of grading joints which ranges from non-existing to severe osteo-arthritis that can be applied to clubfoot radiographs [11]. A rating system evaluates the joints of the foot and grades the degree of arthritis present. Adults with clubfoot demonstrate higher degrees of arthritis which correlates with pain and loss of function [12].

Outcome Evaluations The original Turco Classification can be used to evaluate the overall condition of the foot after treatment [13]. It provides a basic description of the clubfoot correction from excellent, good, fair to failure. In general this classification is quite subjective and insufficient to quantify the results. Laaveg and Ponseti designed a rating system of functional results following treatment with a score of 100 points indicating a normal foot [4]. This includes 30 points for pain; 20 points each for level of activity and patient satisfaction; and 10 points each for motion of the ankle and foot, position of the heel during stance and gait. The International Clubfoot Study Group (ICFSG) has developed an ICFSG outcome evaluation system that combines assessments of foot morphology, function and radiographs [14]. The morphology of the hindfoot, midfoot and forefoot as well as the global morphology of the foot and lower limbs is scored. There are also functional scores for passive motion, muscle function, dynamic function (gait), and pain. The radiographic evaluation scores standing AP, lateral and ankle views for normalcy. Maximum score of 60 indicative the worst results. The American Orthopaedic Foot and Ankle Society (AOFAS) Hindfoot and Midfoot scales are additional clinical rating systems that take into account static, dynamic and quality of life measures. Both scales score pain, function (limitations, gait, foot motion, etc.) and static alignment [15].

Gait Analysis

Gait analysis is a commonly utilized method of measuring function in clubfoot patients over the age 3 years of age. It provides quantitative data on the motion of all of the lower extremity joints (hip, knee, and ankle), temporal spatial parameters (walking speed, cadence, step/stride length, etc.) as well as joint kinetics and electromyography during walking. This data can be compared to age matched controls to identify segmental and planar gait deviations at the pelvis, hip, knee and ankle. Figure 4 is the graphs representing gait kinematics and kinetics of the ankle joint. It is noted the reduction in overall motion at the ankle in the sagittal plane with reduced peak dorsiflexion and reduced peak plantarflexion. The ankle moment is abnormal reduced peak power generation. Also note that it is common for a subject with clubfeet to have less functional ROM during gait then available ROM as measured from the physical examination. Conventional gait modeling identifies the foot as a single segment rotating about a revolute ankle joint. To examine dynamic foot function more appropriately a foot model can be used to measure segmental (tibia, hindfoot, forefoot, and hallux) kinematics. Foot motion analysis allows for more in-depth examination of the segments affected by clubfoot correction and treatment. There are several marker based models that have been used to quantify segmental foot motion [16, 17]. The Milwaukee Foot Model uses weight bearing radiographs to reference the motion of four segments of the foot to the underlying bony anatomy providing offsets that take into consideration any bony alignment deformities [18]. Figure 5 represented segmental motion of the right hindfoot and forefoot of an individual with surgically treated clubfoot. We can note the plantarflexed and inverted hindfoot and the dorsiflexed and adducted forefoot.

Pedobarography analyzes the pressure that is distributed across the plantar surface of the foot when it is in contact with the floor. Often assumptions can be made about the foot’s function based on its aesthetics, but pedobarography can provide another perspective on the skeletal segmental alignment. One method of analysis is to trace the center of pressure (COP), or average pressure, throughout the stance phase of the gait cycle and determine how long the COP lies within each segment of the foot (hindfoot, midfoot, forefoot). The Center of Pressure Progression (COPP) can also be traced to determine if it’s path is varus, valgus or normal through each segment [19]. This system can help determine walking patterns such as calcaneus, or equinus as well as the condition of the medial longitudinal arch (Figure 6).

Functional/Quality of Life Questionnaire Three useful questionnaires for subjects with clubfeet are the Short Form 36 (SF-36), Disease Specific Index (DSI) and Foot Function Index (FFI). The SF-36 is a multipurpose, short-form health survey with only 36 questions. It yields an 8-scale profile of functional health and well-being scores as well as psychometrically-based physical and mental health summary measures and a preference-based health utility index. It is a generic measure, as opposed to one that targets a specific age, disease, or treatment group [www.sf-36.org/tools/SF36.shtml]. Results in treated adults have shown differences from typically developing adults in pain and disability. The DSI has 10 questions, each with 4 possible response codes per question. Topics range from foot pain, shoe wear, social acceptance, disabilities, to personal satisfaction. The FFI is a self-administered index that uses a Likert Scale from 0 to 9 to rate pain, level of difficulty and time spent

for various activities. It consists of 23 items divided into 3 sub-scales. This was developed to measure the impact of foot pathology on function in terms of pain, disability and activity restriction.

FUTURE ASSESSMENT TOOLS Fluoroscopic Imaging System for Foot and Ankle Motion Analysis during Walking While static images with CT and MRI are currently available, dynamic motion measurements of the bones of the foot during walking have not been done. A fluoroscopic system could provide unique motion picture “x-rays” of the bones of the foot and ankle during walking. Together with existing gait analysis technologies, it will be possible to accurately track and describe the skeletal foot motion. This information will allow a team of clinicians, surgeons, engineers and rehabilitative professionals to more accurately plan treatment, monitor progress and provide longer term ongoing care to improve the overall biomechanical function and quality of life.

Kinetic Foot Model Rankin et al. describes several future developments of foot modeling including the use of a kinetic foot model that addresses forces and moments between multiple segments of the foot [20]. Preliminary work has been done by MacWilliams et al. [21] but future work will require the development of technology to adequately measure the distribution of shear forces on the plantar surface of the foot.

Finite Element Modeling

With the development of the technologies mentioned above, future research will most certainly include the use of finite element modeling to study the forces acting within a complex foot. The authors are currently aware of three such models. Gefen et al. developed a model that provides detailed information concerning foot anatomy during ambulation [22], and two similar models have been proposed by Cheung et al. [23, 24] .

Acknowledgement: Authors wish to acknowledge the support of the generous grant for this study from U.S. Department of Education, National Institute on Disability and Rehabilitation Research (NIDRR) , Grant # H133G060252

References: 1.

Dietz, F.R., et al., Evaluation of a disease-specific instrument for idiopathic clubfoot outcome. Clin Orthop Relat Res, 2009. 467(5): p. 1256-62.

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Atar, D., et al., Revision surgery in clubfeet. Clin Orthop Relat Res, 1992(283): p. 223-30.

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Catterall, A., A method of assessment of the clubfoot deformity. Clin Orthop Relat Res, 1991(264): p. 48-53.

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Laaveg, S.J. and I.V. Ponseti, Long-term results of treatment of congenital club foot. J Bone Joint Surg Am, 1980. 62(1): p. 23-31.

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Magone, J.B., et al., Comparative review of surgical treatment of the idiopathic clubfoot by three different procedures at Columbus Children's Hospital. J Pediatr Orthop, 1989. 9(1): p. 49-58.

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Dyer, P.J. and N. Davis, The role of the Pirani scoring system in the management of club foot by the Ponseti method. J Bone Joint Surg Br, 2006. 88(8): p. 1082-4.

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van Mulken, J.M., S.K. Bulstra, and N.H. Hoefnagels, Evaluation of the treatment of clubfeet with the Dimeglio score. J Pediatr Orthop, 2001. 21(5): p. 642-7.

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Dimeglio, A., et al., Classification of clubfoot. J Pediatr Orthop B, 1995. 4(2): p. 129-36.

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Kuo, K.N. and P.A. Smith, Correcting residual deformity following clubfoot releases. Clin Orthop Relat Res, 2009. 467(5): p. 1326-33.

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Smith, P.A., et al., Brace evaluation in children with diplegic cerebral palsy with a jump gait pattern. J Bone Joint Surg Am, 2009. 91(2): p. 356-65.

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Dobbs, M.B., R. Nunley, and P.L. Schoenecker, Long-term follow-up of patients with clubfeet treated with extensive soft-tissue release. J Bone Joint Surg Am, 2006. 88(5): p. 986-96.

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Turco, V.J., Resistant congenital club foot--one-stage posteromedial release with internal fixation. A follow-up report of a fifteen-year experience. J Bone Joint Surg Am, 1979. 61(6A): p. 805-14.

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Celebi, L., et al., Bensahel et al. and International Clubfoot Study Group evaluation of treated clubfoot: assessment of interobserver and intraobserver reliability. J Pediatr Orthop B, 2006. 15(1): p. 34-6.

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Toolan, B.C., et al., An evaluation of the use of retrospectively acquired preoperative AOFAS clinical rating scores to assess surgical outcome after elective foot and ankle surgery. Foot Ankle Int, 2001. 22(10): p. 775-8.

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Theologis, T.N., et al., Dynamic foot movement in children treated for congenital talipes equinovarus. J Bone Joint Surg Br, 2003. 85(4): p. 572-7.

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Graf, A., et al., Long-term outcome evaluation in young adults following clubfoot surgical release. J Pediatr Orthop, 2010. 30(4): p. 379-85.

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Johnson, J.E., et al., Hindfoot coronal alignment: a modified radiographic method. Foot Ankle Int, 1999. 20(12): p. 818-25.

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Jameson, E.G., et al., Dynamic pedobarography for children: use of the center of pressure progression. J Pediatr Orthop, 2008. 28(2): p. 254-8.

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Rankine, L., et al., Multisegmental foot modeling: a review. Crit Rev Biomed Eng, 2008. 36(2-3): p. 127-81.

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Figure legends

Figure 1A: A/P and Figure 1B: lateral radiographs of an adult male who underwent a comprehensive surgical release with moderate to severe osteo-arthritis of the subtalar, tibiotalar, calcaneocubiod and talonavicular joints as graded by the Kellgren system.

Figure 2A: A/P and Figure 2B: lateral views of radiographs of a typically developed foot. 1.) Bimalleolar axis 2.) Calcaneal axis 3.) 2nd metatarsal 4.) 1st metatarsal 5.) Hallux (1st phalanx) 6.) Tibia 7.) Calcaneus 8.) 1st metatarsal 9.) Hallux (1st phalanx)

Figure 3: 3A frontal view, 3B back view, 3C lateral view and 3D medial view of photos of an individual with surgically treated bilateral clubfeet. Note the calf atrophy, hindfoot varus and forefoot adductus.

Figure 4: Gait kinematics and kinetics of the ankle. The graphs represent 0-100% of the Gait cycle, with the subject’s left side plotted in blue, right side in red and normal range in gray. The vertical lines represent the subject’s toe off.

Figure 5: Segmental motion of the right hindfoot and forefoot of an individual with surgically treated clubfoot. The dashed line is normal; the solid lines are 3 different gait cycles of the same foot.

Figure 6: Plantar pressures of a normal foot (A) and an individual with surgically treated clubfeet (B and C). The foot is divided into 3 segments by the horizontal black lines; the hindfoot, midfoot and forefoot. The mean value for the Center of Pressure Progression (COPP) is shown be the solid red line. The ±1 and 2 standard deviations for the mean value of the COPP for typically developing children are shown by the vertical solid black lines. (A) Plantar pressure and COPP of a typical adult. (B) Plantar pressure analysis of an adult who underwent comprehensive surgical release for clubfoot as an infant. The COPP is deviated laterally, indicating a varus loading pattern. (C) Plantar pressure of an adult who underwent comprehensive surgical release for clubfoot as an infant. Static standing alignment of the foot demonstrates hindfoot varus with forefoot adduction; however, COPP is within the accepted range of medial-lateral deviation.

*Title Page

Comprehensive Outcome and Functional Evaluation of Clubfoot Treatment Adam Graf, MS1, Kuan-Wen Wu, MD2, Peter A Smith, MD1, Ken N Kuo, MD2,,3

1

Shriners Hospital for Children, Chicago 2

3

National Taiwan University Hospital

College of Medicine, Taipei Medical University

Corresponding author Ken N Kuo, MD College of Medicine Taipei Medical University 350 Wu-Hsing Street Taipei 110, Taiwan Tel: 886 2 2736 1661 Fax: 886 2 2739 0214 e-mail address: [email protected]

Cover Letter

Professor Ashok Johari Editor-in-Chief Journal of Pediatric Orthopaedic – B

Dear Dr. Johari:

March 1, 2011

This is a manuscript for special issue in memory of Henri Bensahel on clubfoot. It is one of the articles by Henri’s friends who are interested in clubfoot management. This is the work of authors, and there is no conflict of interest. I hope the article is acceptable to the journal standard. I am looking forward to your favorite review. Sincerely yours,

Ken N Kuo, MD National Taiwan University Hospital Taipei Medical University

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Table

Lateral

A/P

Table 1: Radiographic Foot Measurements of a group of Controls and one adult with bilateral clubfeet.

Joint Angle (Degrees) 1st Metatarsalphalangeal Calcaneus-2nd Metatarsal Bimaleolar Axis-Calcaneus Tibia-Calcaneus 1st Metatarsalphalangeal Calcaneus-1st Metatarsal

Controls (n=15) 6.9° -7.4° 6.9° 19.8° 8.9° -39.4°

Clubfoot L R -9° -5° -25° -16° -6° 0° 10° -3° 15° 12° -23° -7°

Table

Table 2: Example Physical Exam Worksheet: Subject with Bilateral Clubfeet Measurement Calf Circumference (cm) Leg Length (cm) Foot Length (cm) Ankle Motion (prone) Dorsiflexion (knee ext) Dorsiflexion (knee flx) Plantarflexion (knee ext) Inversion (knee flx) Eversion (knee flx) Thigh Foot Angle (knee flx) Weight Bearing Observations Hindfoot Midfoot Entire Foot

Left 28 94.5 25.3

Right 32 93 25.3

0° 0° 0° 0° 0° 8° Internal

0° 5° 12° 23° 0° 30° External

Varus/Valgus/Neutral Equinus/Calcaneus/Neutral Supination/Pronation/Neutral Adduction/Abduction/Neutral Internal/External/Neutral Pes Cavus/Flat foot/Neutral

Varus/Valgus/Neutral Equinus/Calcaneus/Neutral Supination/Pronation/Neutral Adduction/Abduction/Neutral Interal/External/Neutral Pes Cavus/Flat foot/Neutral