Journal of Bodywork & Movement Therapies (2014) 18, 4e10

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CASE REPORT

Osteopathic manipulative treatment of congenital talipes equinovarus: A case report Elda Andreoli, M.D. a,*, Andrea Troiani, D.O. b,c, Valentina Tucci, D.O. b,c, Gina Barlafante, M.D., D.O. b,c, Francesco Cerritelli, MPH, D.O. b,c, Gianfranco Pizzolorusso, D.O. b,c, Cinzia Renzetti, M.D., D.O. b,c, Daniele Vanni, M.D. a, Andrea Pantalone, M.D. a, Vincenzo Salini, M.D a a

Postgraduate School of Orthopaedics and Traumatology Department, University G. d’Annunzio, Via dei Vestini, 31, 66100 Chieti, Italy b AIOT Research Institute, Pescara, Italy c European Institute for Evidence Based Osteopathic Medicine (EBOM), Chieti, Italy Received 7 October 2012; received in revised form 1 March 2013; accepted 20 March 2013

KEYWORDS Osteopathic manipulative treatment; Congenital talipes equinovarus; Clubfoot; Foot; Newborn; Manipulation; Conservative treatment; Complementary and alternative treatment

Summary Background: Treatment recommendations for congenital talipes equinovarus are focused on conservative treatments. Objectives: The aim of this paper is to present a case report of bilateral congenital talipes equinovarus treated with two short-leg serial casting in combination with osteopathic manipulative treatment. Methods: A newborn, 12 days old, with severe bilateral congenital talipes equinovarus entered to the Department of Orthopedics at the University of Chieti, Italy. The pediatric orthopedic surgeon applied two single series of short-leg casts, at 12 and 20 days of age. The osteopath scheduled 4 indirect myofascial release techniques sessions. Results: At day 33 the newborn achieved a complete correction of the congenital talipes equinovarus deformity and there was no need to apply a third series of casts. Conclusions: Results from this single case study create an interesting and clear precedent for considering OMT in future clinical trials. ª 2013 Elsevier Ltd. All rights reserved.

* Corresponding author. Tel.: þ39 3396270177; fax: þ39 0854172587. E-mail address: [email protected] (E. Andreoli). 1360-8592/$ - see front matter ª 2013 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jbmt.2013.03.011

Osteopathic manipulative treatment of congenital talipes equinovarus

Introduction Congenital talipes equinovarus (CTE) is defined as a congenital deformity involving one foot or both feet (Batchelor, 1946). It represents one of the most common muscle-skeletal defects at birth and has a prevalence of 1/1000 among live newborns (Dobbs and Gurnett, 2009). Anatomic abnormalities of CTE affect bones and soft tissues, leading to malalignment of the bones and joints of the foot and ankle. It is distinguished from positional foot anomalies for being supinated and adducted, with varism and equinism associated (Song et al., 1999). In many cases the cause of CTE is idiopathic (WynneDavies, 1964), but the deformity has been also found in 20% of newborns with chromosomal abnormalities, arthrogryposis and myelomeningocele (Brewer et al., 1998; Gurnett et al., 2008a). Many theories have been advanced to explain the pathogenesis (Merrill et al., 2011; Feldbrin et al., 1995; Herceg et al., 2006), suggesting that CTE can be etiologically heterogenous or secondary to a primary factor leading to the anatomic abnormalities at the level of the foot. In addition to this, among the most recent theories there is data supporting the genetic basis of CTE (Gurnett et al., 2008b; Alvarado et al., 2011). Although CTE is easy to recognize at birth, it’s severity needs to be assessed depending on the grade of resistance of the foot to manipulation. At first visits, orthopedic clinicians refer to two major systems of classification developed by Dimeglio et al. (1995) and Pirani (2004). Both classifications use scores for several physical findings, giving a total score that correlates with CTE severity. Treatment recommendations for CTE are focused on reducing or eliminating the anatomical deformities, with the patient having optimal mobility and a functional, painfree, plantigrade foot. Most pediatric orthopedic surgeons agree that the initial treatment should be nonsurgical and start as soon as possible after birth (Sætersdal et al., 2012). Nonoperative treatments include several techniques; among these are the Dimeglio (Bensahel et al., 1990a) and Ponseti (1996) methods. The Dimeglio method, also known as the French method, consists of an intensive physiotherapy program with the CTE treated daily with manipulative stretching followed by taping of the leg and foot to a splint. The aim of the physiotherapist is to soften the tissues and to make the foot more compliant (Bensahel et al., 1990b; Dimeglio et al., 1996a). Dimeglio in 1996 reported a success rate of 74% patients free from surgical interventions (Dimeglio et al., 1996b), while others reported a higher rate of children needing soft-tissue release surgery for residual CTE (Richards et al., 2008). The Ponseti method is based on weekly interventions and the technique consists of three main parts: serial manipulation, casting (with or without Achilles tenotomy) and the application of an orthosis. The orthosis is used to position the foot in abduction to maintain the achieved correction (Scher, 2006). Among various treatment protocols concerning CTE deformity, the Ponseti method is the most effective treatment (Matos & de Oliveira, 2010), with 90% a success rate of 90% at short term and long term results being

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equally impressive (Laaveg and Ponseti, 1980; Cooper and Dietz, 1995; Segev et al., 2005). If conservative measures fail to correct CTE deformity, surgery represents the remaining recourse. As a result of nonoperative treatments, the overall number of surgical soft tissue releases has decreased dramatically, with a very low rate of CTE undergoing surgery because of recurrence (Dobbs et al., 2004). However, it is difficult to draw certainty among all these procedures since no structured research has been conducted evaluating the effectiveness of these manipulative techniques. As far as complementary and alternative medicine is concerned, the literature does not provide any evidence of the applicability of these approaches to CTE and consequently no data are available. The aim of this paper is to present a case report of bilateral CTE treated with two short-leg serial casting in combination with osteopathic manipulative treatment (OMT) as a complementary procedure.

Methods Case report A newborn, 12 days old, white Caucasian female with bilateral CTE entered to the Department of Orthopedics at the University of Chieti, Italy. The child was born at term, 39 weeks of gestational age, with vaginal delivery. No fetus complications during the pregnancy were observed. The growth of the fetus was regular along the entire period, following the 50th percentile, and no associated pathologies were diagnosed at birth. The mother, 35 years old, white Caucasian at her third pregnancy, smoker, non-alcoholic, with no history of genetic/congenital disorders, started vasosuprina for premature uterine contractions at 20 weeks of gestational age. The father, 33 years old, white Caucasian presented no congenital/genetic disorders as well as no history of acquired diseases.

Figure 1 Picture documenting the bilateral CTE before casting. Age of the child: 12 days.

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E. Andreoli et al.  adduction of the forefoot relative to the hindfoot in the horizontal plane: 3 (right foot); 3 (left foot).

Assessment and orthopedic treatment The newborn underwent the first pediatric orthopedic visit at the age of 12 days (Figure 1). At the initial presentation of the patient to the unit, the orthopedic surgeon assessed a complete history of the child and measured the passive range of motion of dorsiflexion, plantar flexion, inversion, eversion and determined rear foot varus angle and forefoot adduction angle. The severity of CTE deformity was assessed using the Dimeglio classification system (Dimeglio et al., 1995) shown in Table 1. According to the Dimeglio grading system and with the use of a handheld goniometer, the pediatric orthopedic clinician assigned a score (0e4) to each of the following parameters:  equinus deviation in the sagittal plane: 3 (right foot); 4 (left foot)  varus deviation in the frontal plane: 3 (right foot); 3 (left foot)  derotation of the calcaneopedal block in the horizontal plane: 3 (right foot); 3 (left foot)

Table 1

An additional point for a medial crease was added for each foot, producing an overall deformity score of 13 and 14 points in relation to the right and left foot, respectively. According to the severity of the deformity, the feet were classified as grade severe feet (11e15 points). No X-rays were taken to check for structural abnormalities, neither interobserver or intraobserver error was evaluated. After the physical examination, the surgeon applied a bilateral short-leg casts to the area below the knee to the foot. Casting was applied with minimal force, not requiring anesthesia, preventing the deformity from worsening and to take advantage of the neonate’s potential for remodeling. The child was cast for eight days. At day 20 the feet had achieved satisfactory initial correction and the second series of short-leg casts was applied with minimal force. At that time the goniometer re-evaluation was not performed to quantify the achieved correction. The third orthopedic visit was planned at day 33 (Figure 2).

Description of the Dimeglio classification system.

Date_____________ Name______________________________________ Current Cast Number (circle):

Foot (circle):

R

L

Atypical (circle if yes)

0 1 2 3 4 5 6 7 8 9 ______

Complications (circle)

0)None 1)Rocker sole 2)Maceration 3)Abrasion 4)Blister 5)Slough 6)decubitus 7)Cast saw injury 8)Cast intolerance/removal 9)Cast fell off 10)Other_______________________

Surgical Date:

Procedure: (circle)

____________

4)PMR

Date DBB Applied: ____________

1)None 2)Per-Q Achilles tenotomy 3)Open TAL/post release 5)Anterior tibialis transfer 6)Other ______________

Compliance:

1)YES

2)NO

Wearing:1)full time

2)Night/Naptime

Age bar stopped at:_____yrs

Stopped by:

_____mos

1)MD

2)parents Dimeglio/Bensabel 1. Equinus Dorsiflexion

Points _________________°

Points

For parts 5-8, Mark points as, Present=1, Absent=0

3. Midfoot rotation (Horizontal plane)

Plantarflexion 45°- 90°

4

Supination 45°- 90°

4

5. Posterior Crease

Plantarflexion 20°- 45°

3

Supination 20°- 45°

3

6. Medial Crease

Plantarflexion 0° - 20°

2

Supination 0°- 20°

2

7. Cavus

Dorsiflexion 20° - 0°

1

Pronation 20°- 0°

1

Dorsiflexion > 20°

0

Pronation > 20°

0

2. Hindfoot varus

8. Abnormal underlying muscolature

4. Forefoot adduction (on hindfoot)

Varus 45° - 90°

4

Adductus 45° - 90°

4

TOTAL SCORE

Varus 20° - 45°

3

Adductus 20° - 45°

3

Type I: 0 - 5 points

Varus 0° - 20°

2

Adductus 0° - 20°

2

Type IIa: 6 - 10 points

Valgus 20° - 0°

1

Abductus 20° - 0°

1

Type IIb: 11 - 15 points

Valgus > 20°

0

Abductus >20°

0

Type III: 16 - 20 points

Points

Osteopathic manipulative treatment of congenital talipes equinovarus

Figure 2 Picture taken at the end of the osteopathic and orthopedic treatments. Age of the child: 33 days.

Osteopathic manipulative treatment Osteopathic medicine is a form of drug-free non-invasive manual medicine, designated as complementary and alternative medicine (CAM). Osteopathic practitioners aim to use their understanding of the relationship between structure and function to optimize the body’s self-regulating, self-healing capabilities. Two essential components of osteopathic health care are the structural evaluation of the patient for diagnosis and an array of manipulative techniques for treatment (www.woho.org, 2008). The aim of the structural examination is to locate somatic disfunction (SD), defined in the osteopathic literature as impaired or altered function of related components of the somatic system: skeletal, arthrodial and myofascial structures; and related vascular, lymphatic, and neural elements (www.aacom.org, 2002). In newborns the structural exam is usually performed with the child lying down on the table. Diagnostic criteria for SD are focused on tissue texture abnormalities and tone. Areas of asymmetry and misalignment of bony landmarks are evaluated. The quality of motion, its balance and organization are noted. In treating children during the very first days of life osteopathic practitioners use a wide variety of therapeutic manual techniques to increase range of motion, to improve physiological function and/or support homeostasis that has been altered by SD. The term osteopathic manipulative treatment (OMT) currently encompasses more than twenty types of osteopath-performed manual treatments. The choice of OMT technique is secondary to the physical condition and age of the patient, and depends upon the osteopath’s experience with various approaches (Kappler et al., 1997). The osteopathic evaluation of the newborn presented in the present case study led to a precise and individual plan for her treatment. Before the initial treatment, both parents were asked to sign an informed consent form and four osteopathic treatments were scheduled as follows: two treatments at the age of 5 and 10 days plus two treatments

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at days 17 and 24, the latter with the child being in a bilateral casts. Indirect myofascial release technique (indirMFR) was the OMT technique of choice. This choice was based on multiple factors, such as: age of the patient, safety, physical condition (in the last treatments the patient was cast), experience and expertise with myofascial release technique (MFR). The sequence, dose and frequency of treatment were left to the discretion of the osteopaths and not based on a predetermined protocol. The specific MFR techniques used in the four osteopathic sessions were addressed to SD of the following structures: upper cervical and cranial base, ribs and diaphragm, thoracolumbar spine and sacrum. Both tibiae and ankles were treated only in the first two osteopathic sessions, when both legs were free from casts. The MFR approach is a system of diagnosis and treatment added to the armamentarium of osteopathic medicine by its founder Dr Andrew Taylor Still, which uses continual palpatory feedback to achieve release of myofascial tissues (Glossary of Osteopathic Terminology, 2009). In indirMRTs the dysfunctional tissues are moved away from the restrictive barrier (a functional limit that abnormally diminishes the normal physiologic range of motion) until tissue tension is equal in one or all planes and directions (Glossary of Osteopathic Terminology, 2009). This is accomplished by relaxing contracted muscles, increasing blood flow and lymphatic drainage in the area (DiGiovanna et al., 2004), allowing the body’s inherent ability for self correction to restore the optimal performance of the body.

Results At day 33, when the third orthopedic visit was scheduled, a complete correction of the CTE deformity was achieved (Figure 2). This made unnecessary both the third series of casts and orthosis. The aspect of both lower limbs were normal, there were no further deformities and mobility was reestablished. The achieved correction did not relapse during the first 18 months of follow-up.

Discussion Three-dimensional complexity, and the large number of joints in the foot, play important roles in static and dynamic function. The newborn foot undergoes important morphological changes in the early periods of development and the treatment of neonatal foot pathology has proven to be challenging (Dobbs et al., 2004). CTE is a deformity that has been discussed in the literature since 1946 (Batchelor, 1946), but its assessment and treatment remain poorly understood in many respects. Uncertainties about the CTE etiology (Merrill et al., 2011; Feldbrin et al., 1995; Herceg et al., 2006; Gurnett et al., 2008b; Alvarado et al., 2011) and lack of a single classification system (Dime ´glio et al., 1995; Bensahel et al., 1990a; Pirani, 2004), make determination of a specific protocol treatment difficult. As far as CTE assessment, no method of classification is widely accepted. The Dimeglio and Pirani classification

8 systems have the best resonance and therefore are used by the majority of pediatric orthopedic surgeons (Pirani, 2004). Several treatment programs (Chu et al., 2010) have been reported in the literature, consisting of intense physical therapy sessions, taping, serial casting, orthosis, as well as the involvement of parents and caregivers to learn the techniques so as to carry out the therapy at home. In case of failure, surgery remains a valid option, but rather complementary and not alternative to conservative treatments (Dobbs et al., 2004). The relative effectiveness of different treatment methods is far from clear in terms of long-run outcomes, stability of results and risk of recurrences (Porecha et al., 2011). However, the most effective and common treatment used is the Ponseti method (Matos & de Oliveira, 2010; Bridgens and Kiely, 2010), with very few relapses and side effects, either short term or long term (Cooper and Dietz, 1995; Abdelgawad et al., 2007). The Ponseti method is a specific method of manipulations associated with serial long leg casting, from 3 to 8 (Herzenberg et al., 2002; Porecha et al., 2011), and a percutaneous Achilles tendon tenotomy release in severe cases. The treatment must be started within the first week of life and casting renewal every 5e7 days. In most cases, after the fourth cast renewal, all the deformities are corrected, with the exception of the hindfoot equinus deformity. In such cases, a corrective Achilles tendon tenotomy is performed. A final long leg cast is applied after the tenotomy, and the foot is positioned in abduction (70 ) and dorsiflexion (5 e10 ). After the final cast is removed, a foot abduction brace is used to prevent relapses (Dobbs and Gurnett, 2009). As far as complementary and alternative methods are concerned, the literature lacks data documenting the effect of non-conventional treatments in infants affected by CTE. The present case report of bilateral CTE has shown that four OMT sessions, together with only two series of bilateral short-leg casts, dramatically decreased the severity of the deformity. At the end of the osteopathic and orthopedic treatments, without any additional medical intervention, a complete correction of the CTE was achieved; after 18month follow up no relapses were recorded. Cases comparable to the newborn presented in this study, with a remarkable morphological alteration of the feet (score of 13 points for the right foot and 14 points for the left foot, according to the Dimeglio classification system) would probably need a median treatment period of 6 weeks, followed by boot application and bar treatment for several months. In the present case report, the CTE was treated by welltrained osteopaths and MFR techniques were the OMT techniques specifically chosen for the treatment. These techniques were employed to facilitate the release of fascial restrictions palpated through the skin of the infant. If the anatomic relationship between muscles, bones, vessels and viscera is considered, fascia represents the system surrounding and supporting all components of the body. From this prospective, fascial restrictions at any level of the body may affect any structure enveloped by fascia (Greenman, 1989), with stress on the surrounding soft tissues at mechanical and physiological levels (Tozzi et al., 2012). In the newborn presented in this case study, the application of MFR techniques was aimed to release tensions within the tissues,

E. Andreoli et al. increase the range of motion of the foot and ankle, soften the tissues and make the foot more compliant. Most SD diagnoses presented a significant component of fascial restriction, resulting in connective tissue tightening. After the application of MFR, especially in the region of the lumbar spine and pelvis, the practitioner was able to return the foot and its tissues to normal function and position. However, in MRT the soft tissues treated are not only muscles and fascias, but also tendons, ligaments and cartilage (DiGiovanna et al., 2004). For this reason, MFR technique applied to the pelvis were designed to lead to tissue changes in the lower limbs of the newborn. Muscle tension in both legs decreased, as well as fascia shortening, while tendons and joints function and symmetry improved. In the absence of clear evidence explaining the mechanisms underlining the traditional and conservative treatments, it’s difficult to address the specific role of OMT. From an orthopedic perspective, changes in the structure of the collagen, secondary to serial casting, are considered to play an important role in repositioning the bones of the foot (Bridgens and Kiely 2010). On the other hand, MFR techniques performed by osteopaths and manual therapists are known to affect the viscoelasticity of fascia, through a re-organization of the collagen bundles within the tissue (Grinnell, 2008; Swartz et al., 2001). The data available in the osteopathic literature also provide evidence of the effects of indirect OMT in fibroblasts. In 2007, Meltzer and Standley developed an in vitro model involving human fibroblasts and started experiments on strain-regulated fibroblast-derived cytokines. They concluded that indirect OMT techniques modulate the secretion of proinflammatory and anti-inflammatory interleukins. Few years later, Meltzer et al. (2010) investigated the effects of modeled MFR after the application of repetitive motion strain in the same in vitro model. They showed that MFR reverses several morphological changes in fibroblasts e apoptosis and loss of intracellular integrity e that occur after repetitive motion injury. Given this evidence, we propose that the modulation of the inflammatory and neuroendocrine responses, along with tissues texture changes, increased elasticity and improved range of motion, and that this may be the mechanisms by which OMT decreased the severity of CTE deformity. Among various treatment methods concerning CTE, the complementary use of OMT has been reported to be of great importance in the treatment of the newborn presented in this report. No side effects were recorded, neither was there parental involvement in taking the therapy for home use required. Additionally the OMT sessions were not as intensive as the usual physiotherapy programs, and the rapidity in resolution was equally impressive. This case report calls for further investigations, with RCT looking at the effect of OMT in larger population of newborns affected by CTE.

Conclusions The use of OMT to correct the CTE deformity represented an appealing option in the treatment of the newborn of this case report.

Osteopathic manipulative treatment of congenital talipes equinovarus Results from this single case study create an interesting and clear precedent for considering OMT in future clinical trials.

Conflict of interest Authors declared no conflicts.

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Osteopathic manipulative treatment of congenital talipes equinovarus: a case report.

Treatment recommendations for congenital talipes equinovarus are focused on conservative treatments...
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