Treatment of flexion contracture of the knee joint in children with arthrogryposis: A literature review

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Abstract


Knee joint lesions are observed in 58%-90% of patients with arthrogryposis. Majority of these cases involved flexion contracture of the knee. Ambulation is possible with a deficit of knee extension of 15-20°. Currently there is no standardized treatment in global practice, which makes the treatment of this disease extremely challenging. Clinicians must take into account the degree of flexion contracture, segmental level of the spinal cord lesion, and the age of the child when considering treatment options. In this study, national and international literature relating to the clinical profile, classification, and conservative and surgical treatment of this condition were reviewed. The following treatments have been considered for this condition: casts, extensor osteotomy, arthrolysis of the knee, the use of external fixation devices, and hemi-epiphysiodesis. The high probability of relapse for flexion contracture of the knee joint in children with arthrogryposis has been described.

Arthrogryposis is a disease characterized by congenital contractures of two or more joints in nonadjacent regions, combined with muscular hypotrophy or atrophy and signs of motor neuron lesions in the spinal cord; such a diagnosis is made after other known systemic diseases have been excluded [1-3].

In 1573, Pare first described a sick child with deformities of the limbs similar to those in patients with arthrogryposis in his paper “Monster and Wonders” [4]. Then, in 1841, Otto outlined arthrogryposis as a syndrome of multiple congenital contractures in “Monstrum Humanum Trunco Nimis Brevi et Extremitatibus Incurvatis.” It was assumed that the cause of the contractures was joint deformation. In 1889, Phocas separated and crossed all the shortened muscles around the knee joint in a patient with arthrogryposis and was amazed with the stability of the contractures. Based on these data, in 1913, Rhocher proposed to use the term “multiple congenital rigid joints.” Only later, in 1923, did Stern coin the term “congenital multiple arthrogryposis,” and this has since been the term used for this condition [4, 5].

According to some authors, the incidence of the specified pathology is 1 in 3000-5100 newborns [3, 6-8]. The knee joints in arthrogryposis are affected in 58%-90% of the cases [9-12], and the most common lesions are flexion contractures. Histological examination of the nervous system tissues with arthrogryposis reveals degeneration of the motor neurons in the anterior horns of the spinal cord [13]. Brown et al. (1980) attributed the injury of the anterior horns in the cervical and lumbar parts of the spinal cord in arthrogryposis to multiple deformations of the limbs, emphasizing eight types. Deformation of the knee joints are observed in the 3rd type (spinal cord lesion at L4, L5, and S1) as extensor contractions, in the 4th type (lesion at L3, L4, and partially at L5) as flexion contractures, and in the 5th type (lesion at L3, L4, and partially at S1-S2) as extensor contractures with valgus deformation of the knee joints [14]. With respect to severity, Staheli et al (2008) classified the flexion contractures in the knee joints based on the angle of deformation as very mild (0°-20°), mild (20°-40°), moderate (40°-60°), severe (60°-80°), and very severe (over 80°) [15].

Flexion contractures of the knee joints restrict walking and are extremely difficult to treat [10, 16-18]. It has been shown that flexion contractures up to 20° does not substantially prevent walking and allows the use of orthoses in patients [15]. However, such contractures tend to increase energy consumption during movement, shorten the step length, and increase oxygen consumption, which negatively affects gait and can result in falls [19]. Contractures with an extension deficit of 20°-40° complicate the use of orthoses, interfere with walking, and usually require surgical treatment [20].

Böhm et al noted that flexion contractures of the knee joints affected gait, and they have conducted a study that associated this pathology with abnormal movements of the trunk during walking [21]. A biomechanical study was conducted in 18 children with congenital multiple arthrogryposis, where the authors analyzed three-dimensional motions of the pelvis and thorax and deviation of the spinal axis during walking. The study noted a correlation between decreased muscle strength, degree of contractures in the lower extremities, and severity of deviations in the thoracic and/or lumbar spine. Therefore, the authors noted the importance of considering these parameters in contracture treatment to avoid deformities of the spine [21]. In addition, in flexion contractures the gravity center is shifted posteriorly; which increases the load on the hip extensor and knee joints and results in faster fatigue as well as the development of osteoarthritis with increased age [12].

A study was conducted in Shriners Hospital for Crippled Children, Tampa Unit, Tampa, Florida, USA, where the knee joints of 46 children with congenital multiple arthrogryposis were assessed. The children’s ages ranged from 2 months to 21 years (mean 7.2 years), and they all had flexion contractures of the knee joints. Changes in the patella and its position were detected in up to 68% of cases and included the following: dislocation, shortening of the patellar ligament, underdevelopment, impairment of ossification, absence or change in its shape, and fractures. Degenerative changes in the periarticular tissues, such as thickening of the periarticular tissue, fibrofatty degeneration, thickening and shortening of the knee joint capsule, and ligament instability, were observed in 59% of cases. Attention was drawn to radiographic findings such as changes in the shape and size of the tibia and femur, changes on articular surfaces, and fractures during conservative treatment as well as osteoporosis [22].

Evaluation of the lower limb muscles of children with knee joint flexion contractures in arthrogryposis revealed hypoplasia, quadriceps atrophy as well as changes in the membrane potential of the muscles on the back of the thigh [12].

According to Lampasi et al (2012), conservative treatment should be started in the neonatal period in the form of exercises aimed at stretching the muscles and tendons, positioning, and the use of removable joint-immobilizers [12]. Guidera and Kortright are proponents of early surgical intervention on the knee joint in children with flexion contractures in arthrogryposis to avoid the development of periarticular tissue and joint remodeling [22].

Studies on the treatment of this type of deformation encountered in literature are largely descriptive. Lloyd-Roberts et al. (1970) applied the method of staged cast treatment of flexion contractures of the knee joints in 15 patients, and six of them have achieved correction of the contracture with good long-term outcomes. The remaining nine patients showed suboptimal and transient effect of such treatment [23].

After studying the results of treatment of patients with arthrogryposis, the authors concluded that conservative treatment with stage plaster bandages in majority of the cases eliminated flexion contractures of the knee joints as long as the angle did not exceed 150° because this method had a limitation in the ability for deformation treatment [12, 24].

There is no consensus in literature regarding the timing of surgical treatment in patients with flexion contractures of the knee joints in arthrogryposis. If conservative treatment fails, some authors recommend surgery at the age of 10-12 months, considering the normal range of motion in ankle and hip joints. In the presence of concomitant pathology of the adjacent joints on the lower limbs, following this algorithm has been suggested for starting the treatment: up to 3-4 months for the treatment of foot deformities, 6-8 months for flexion contractures in knee joints, and 8-12 months for hip joint deformities [12]. Surgical treatment is indicated for flexion contractures of the knee joints with angles of more than 150° [25].

In mild forms, surgical operations on the soft tissue, lengthening of the tibia flexors, and posterior capsulotomy of the knee joint are effective [17, 26, 27]. 

After this intervention, it is recommended to refrain from limb fixation with plaster bandage in the position of full contracture correction to avoid simultaneous stretching of the neurovascular bundle. Gradual treatment of the contracture with plaster bandages and plasterotomy on the posterior surface of the knee joint is preferred during the postoperative period. The rate of recurrence after this method of treatment was 26%-32% [12].

In cases of rigid, recurrent contractures of more than 120°, supracondylar extension osteotomy of the femur is recommended, complemented with lengthening of the tibia flexors [28]. Osteotomy reduces the risk of neurovascular disorders in the postoperative period by shortening the femur. The likelihood of recurrent flexion contracture is high when this surgery is delayed until adolescence [4, 26, 29]. According to some authors, the frequency of recurrent flexion contractures after femur osteotomy reaches 50% in the first 48 months after surgery. Deformity recurrence was observed in 19 postoperative patients (37 osteotomies) after 29 interventions, i.e., in 78.4% patients. The average rate of angle recurrence was 0.69° per month [20]. Therefore, other authors recommend performing a femur extensor osteotomy in older children who do not have knee joint extension of more than 40°. They describe both wedge-shaped, triangular shortening femur osteotomy and perforation of the anterior cortex of the distal femur, combined with the release of the knee joint soft tissues to reduce the degree of femur shortening [12, 30].

The implementation of femur extensor osteotomy restricts the flexion in the knee joint which may hinder the ability to sit and move in transport vehicles [31]. In addition, this operation results in the deformation of the distal femur, which is a cosmetic defect.

A study on rats was conducted in the University of Ottawa with the experimental creation of flexion contractures of the knee joints. The study results allowed authors to prove that time without treatment of this pathology is directly proportional to the number of major and irreversible structural changes in the joint [19]. Therefore, Delbello and Watts believe that there is no reason to delay starting treatment until an older age when the risk of recurrence is lower. They suggest that early restoration of the child’s ability to walk and improvement of the child’s social adaptation are more important than the risk of recurrence after surgical treatment [27].

The issue with recurrence in flexion contracture treatment with conservative methods is extremely important [11, 32, 33]. According to various authors, the recurrence rate varies from 50% [34] to 100% [35]. Lack of prevention methods for the formation of flexion contractures after treatment also leads to rapid recurrence [24, 36]. In general, the need for early implementation of extension osteotomies in patients with flexion contractures of the knee joints is still debatable [23, 24, 28].

Lampasi et al. (2012) describe the clinical case of a child with arthrogryposis who had bilateral dislocation of the hips and flexion contractures of the knee and clubfoot. She underwent surgical release of the knee joint from both sides, followed by open reduction of the hips in 10-12 months, and also underwent conservative treatment of clubfoot. At 7 years, she experienced a recurrence of the contracture at the level of the left knee which was an indication for extensor femur osteotomy on the left. At 11 years, there was a recurrence of knee joint flexion contracture. At 30 years, this woman had full extension in the knee joints; however, the amplitude of active movements in the left knee joint was only 20°. Pain in the joint was also periodically noted [12].

Lampasi et al. (2012) described the treatment of a 12-year-old boy with congenital multiple arthrogryposis who underwent flexion contracture treatment through a release of the knee joint at the 1 year, followed by femur osteotomy at 6 years. After this treatment, there was a deficit in the extension of the knee joints of 20°, which allowed the use of an orthosis. Further treatment of contractures was planned after growth [12].

The application of the Ilizarov apparatus with simultaneous intervention on the soft tissues in severe contractures of the knee joints and feet allows elimination of deformation in two limb segments at the same time [36-38]. According to other authors, this method is indicated for contractures of 40°-60° [12]. As an independent method of treatment, a distraction apparatus was used in children with flexion contractures in the knee joints with the correct inter-relations of the femur and tibia in the frontal plane (no valgus and varus) [24, 40, 41]. The treatment of flexion contractures with an external fixation device required the addition of a posterior arthrolysis on the knee joint to prevent the recurrence of flexion contracture secondary to retraction of the stretched tissues [1, 24, 37].

Using an external fixation device in patients with a mean contracture degree of 72.5°, Van Bosse et al (2007) achieved a complete correction of the knee joint flexion contracture in all patients in an average of 102 days, with a correction rate of 1-2 mm per day from the 3rd day after applying the device [41]. However, this technique has a number of complications, such as fractures and slipped femoral epiphyses secondary to osteoporotic changes during prolonged distraction, subluxation of the tibia, infection, neurovascular disorders, and deformation recurrence [12].

In patients with severe quadriceps hypoplasia, surgical treatment is performed in two stages. The first stage is performed to eliminate knee joint flexion contractures. The second stage follows in 2-3 months, when the flexor muscles of the lower leg are transplanted to the quadriceps [37]. In recent years, new methods have been developed for the treatment of knee joint flexion contractures, including temporary epiphysiodesis of the distal femur growth plate at the anterior surface using a figure-of-eight plate [25, 42, 43]. According to Kramer and Stevens (2001), the degree of contractures can be corrected on average of 20°, provided the ongoing skeletal growth [11, 42].

Deformation may be reduced by correcting limb contractures with the maximum use of the growth plate potential, and as a result, interventions, such as osteotomy, can be delayed or completely eliminated [44]. A number of authors recommend performing hemiepiphysiodesis as early as possible because it is associated with the improvement of the patient’s quality of life.

There are several publications reporting the benefits of hemiepiphysiodesis over other techniques. This technique is minimally invasive, does not require postoperative immobilization, can be combined with the release of the knee joint soft tissues, and there is no loss of flexion after intervention. This method also has a low incidence of complications [12, 41, 44].

Klatt and Stevens performed a comparison of complications after femur extensor osteotomies and hemiepiphysiodesis with plates on the anterior surface of the distal femoral condyles in children with knee joint flexion contractures in arthrogryposis. They found that the possible bias and inconsistency of the plates and staples in hemiepiphysiodesis are less serious complication compared with complications, such as neurovascular disorders, bleedings, fractures, and recurrence of the deformity, after femur extensor osteotomy. They recommend implantation of two figure-of-eight plates for contracture correction from both sides at the anterior surface of the distal femur [30, 41]. Parikh and Crawford (2004) published the results of a study in which figure-of-eight plates were installed in 16 children with knee joint flexion contractures to the anterior femur surface in arthrogryposis. The average age of patients at the time of surgery was 7.4 years (range, 4.3-11.8 years). In the next 2-3 years after surgery, the correction amounted to an average of 18.4°. They recommended this treatment for contractures less than 45° [45].

Janfranchi and Masquijo (2015) studied the treatment results in 18 patients (29 knee joints) after temporary hemiepiphysiodesis was performed for flexion contractures of knee joints in arthrogryposis. Complete correction of the flexion contracture of the knee joint was achieved in 100% of cases. This technique is an alternative to other types of interventions, such as arthrolysis of the knee joints, use of external fixation devices, and extensor osteotomies [44].

This literature review shows that various conservative and surgical methods of treatment are currently used for the treatment of flexion contractures of the knee in children with arthrogryposis, but their degree of efficacy is not high enough. In recent years, temporary hemiepiphysiodesis on the anterior surface of the knee joint has been considered as the advanced method and is mainly used in mild degrees of flexion contracture. Arthrolysis of the knee joint with the use of external fixation devices is primarily used for moderate and severe degrees of flexion contractures. However, there is a lack of a unified approach in determining age indications and the choice of the most appropriate methods of conservative and surgical treatment according to the severity of flexion knee joint contractures.

Funding information and conflicts of interest

The work was performed with the support of Turner Scientific and Research Institute for Children’s Orthopedics. The authors declare no potential conflicts of interest related to the publication of this article.

Svetlana A Mulevanova

The Turner Scientific and Research Institute for Children’s Orthopedics

Author for correspondence.
Email: struna117@mail.ru

Russian Federation MD, PhD student of the department of arthrogryposis. The Turner Scientific and Research Institute for Children’s Orthopedics.

Olga E Agranovich

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: olga_agranovich@yahoo.com

Russian Federation MD, PhD, professor, head of the department of arthrogryposis.The Turner Scientific and Research Institute for Children’s Orthopedics.

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