Treatment of wrist deformities in children with arthrogryposis multiplex congenita

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Abstract


Introduction: Treatment of wrist contractures in children with arthrogryposis multiplex congenita (AMC) is extremely problematic because of the high incidence of recurrence. This study aimed to improve the outcome of wrist contracture treatment in children with AMC.

Materials and Methods: A total of 90 patients (162 wrists) were examined and treated. Patients were assessed using a number of clinical, radiological, and electrophysiological examinations. There are several different clinical variants of wrist contracture, including flexion contracture of the wrist, flexion contracture associated with ulnar deviation, and isolated ulnar deviation of the wrist. Patients were divided into three groups according to the level of spinal cord lesion: С6-С7, С5-С8, and С5-Th1. As the number of damaged spinal cord segments increased, the amplitude of passive and active movements, degree of passive correction, muscle power, and wrist function decreased. Surgical treatment involved the following three approaches: tendon transfers, tendon transfers and carpal wedge osteotomy, and tendon transfers with carpal wedge osteotomy and shortened osteotomy of the forearm.

Results: Analysis of treatment results showed that patients with segmental spinal cord lesions at the  С6-С7 and С5-С8 level were mostly associated with a good outcome, whereas patients with lesions at the  С5-Th1 level achieved satisfactory outcomes.

Conclusions: Patients with segmental lesions of the spinal cord at the С6-С7 and С5-С8 level were associated with restoration of active wrist extension up to the neutral position or more and were expected to achieve significant improvement of hand function. Patients with spinal cord lesions at the C5-Th1 level exhibited significant lesions of the muscles, along with bone deformities. Consequently, surgical treatment could only achieve functional wrist position with minimal improvement of hand function. Using differential approaches in the treatment of wrist contracture that are selected by determining the level of spinal cord lesion will enable physicians to predict the outcome and improve the function and appearance of the wrist.


Introduction

Arthrogryposis multiplex congenita (AMC) is a disease characterized by non-progressive contractures of two or more joints with symptoms of anterior horn motor neuron injuries. Deformations of upper limbs in patients with AMC have been found in 95% of cases [1-3]. Self-care is limited in such patients, who need to use both hands or other adaptive mechanisms to perform basic everyday activities.

Wrist joint deformity is the second most common type of musculoskeletal injury in children with AMC [4-6]. The most common injury is flexion contracture in combination with ulnar deviation of the hand [2, 6]. Few classifications of wrist joint deformities in this disease with detailed descriptions of how each type differs are available in the literature [7].

Most authors agree that conservative treatment should be initiated as early as possible after birth and should include daily stretching exercise and physiotherapy (PT) in combination with stage plaster bandage and orthoses [2, 7, 8]. Various surgical options targeting the correction of wrist joint deformations, such as tendon and muscle plasty, allodesis, arthrodesis, carpectomy, shortening osteotomy of the forearm bones, and deformity correction with external fixation, have been proposed [4, 7, 9-14]. However, the treatment of this disease is extremely problematic in this group of patients because of the high incidence of recurrence [4, 7, 15, 16].

Materials and methods

During 2009–2015, we examined and treated 90 patients (162 hands; age range, 1 month–18 years) with AMC and wrist joint deformities. Clinical, radiological, and electrophysiological examinations were performed. The patients were divided into three groups according to clinical and neurological data: patients with a primary injury of the anterior horn motor neurons at the C6-C7 (18 patients, 33 hands), C5-C8 (35 patients, 62 hands), or C5-Th1 (38 patients, 67 hands) spinal segments.

During clinical examination, we determined the type of wrist joint deformation, hand position in the frontal and sagittal planes, range of possible passive correction, amplitude of active and passive movements in the wrist joint, cosmetic appearance, and functional ability of the hand to grip, as well as the presence of associated deformities of the upper limb. The gripping function of the hand was assessed in children older than 1.5 years using the method by Klimont [17]. The four main types of grip, i.e., terminal, lateral, morphogenetic, and boathook, were analyzed with respect to the child’s age and size of the hand using playing tools and toys of different shapes and sizes. Calculation was made according to the formula: GF (%) = [(C1 + C2 + + C3 + C4) × 100]/12, where GF is the functional ability of the hand to grip, C1 is the estimated terminal grip in points, C2 is the estimated lateral grip in points, C3 is the estimated morphogenetic grip in points, and C4 is the estimated boathook grip in points. A GF of 70%–100% indicated a high functional ability of the hand to grip, a GF of 40%–69% indicated a moderate ability, and a GF of <40% indicated a low ability.

Analysis of the wrist joint deformation revealed the following types: flexion contracture of the wrist joint (104 hands, 64%), flexion contracture in combination with ulnar hand deviation (39 hands, 24%), and isolated ulnar hand deviation (19 hands, 12%). In most cases, there was a bilateral symmetric injury of the wrist joint. In an evaluation of the relationship between the type of wrist joint contracture and the level of spinal segmental injury, flexion contracture of the wrist joint was observed in the majority of cases, with equal incidence in all three groups (Fig. 1). Flexion contracture of the wrist joint combined with ulnar deviation was more frequent in the group with injuries of the C5-C8 spinal segments, and isolated ulnar hand deviation was more frequent in the group with the involvement of C6-C7 spinal segments. Cosmetic hand appearance was affected in all patients.

With an increasing number of involved spinal segments, the degree of passive correction for all components of deformation, amplitude of active and passive movements in the wrist joint, and functional ability of the hand to grip are decreased and the degree and frequency of associated deformities of the upper extremities are increased (Figs. 2–4). Thus, there were decreases of 44% and 69% in the amplitude of passive and active movements in the sagittal plane of the wrist joint, respectively, relative to normal values in patients with injuries of the C6-C7 
spinal segments, of 53% and 73%, respectively, in patients with injuries of the C5-C8 spinal segments, and of 69% and 88%, respectively, in patients with injuries of the C5-Th1 spinal segments. Movements sectors were outside the functional range. Active and passive extensions were largely limited relative to flexion in all groups, which indicated a more severe injury of the extensor hand muscles.

The functional ability of the hand to grip before surgery was high (95% ± 4%) in patients with injuries of the C6-C7 spinal segments, moderate (69% ± 18%) in patients with injuries of the C5-C8 segments, and low (23% ± 18%) in patients with injuries of the C5-Th1 segments.

In an evaluation of the presence of associated deformities of the upper extremities in patients of all groups, it was noted that the wrist joint deformity was most frequently associated with adduction–flexion (flexion) contracture of the first finger, as well as with contractures of the triphalangeal fingers. Pronation contractures of the shoulder and forearm were observed in 18% of patients with injuries of the C6-C7 segments, 55% of patients with injuries of the C5-C7 segments, and 100% of patients with injuries of the C5-Th1 
segments.

X-ray examination

X-ray examination showed different types of carpal coalition in 29% of the patients (Fig. 5), and this condition was most prevalent in children with isolated ulnar hand deviation (68%). The fusion of the capitate and hamate bones was the most frequently observed condition (49%). When analyzing the incidence of carpal coalition depending on the level of the segmental injuries, we found that different variants, including the complete fusion of the carpal bones, were noted in the group of patients with injuries of the C5-Th1 spinal segments (43%).

Figure 5. Carpal coalition in patients with deformities of the wrist joint in AMC. A) Fusion of capitate and hamate bones in patient S., 2.5 years old, with injury of the C6-C7 spinal segmental levels. B) Lunate-triangular coalition of patient B., 10 years old, with injury of the C5-C8 spinal segmental levels. C) Bilateral complete fusion of carpal bones in patient K., 12 years old, with injury of the C5-Th1 spinal segmental level

Electrophysiological study

The degree of neuromuscular damage of the upper limbs was possible to assess when performing surface EMG stimulation before surgical treatment. We determined the amplitude and structure of electrogenesis following muscles of the forearm and hand: m. flexor carpi radialis, m. flexor carpi ulnaris, m. flexor digitorum superficialis, m. extensor carpi radialis longus et brevis, m. extensor carpi ulnaris, and m. extensor digitorum communis. The parameters of the EMG amplitude of the respective muscles in the middle-aged patients in the study groups were compared with those in similarly aged patients without pathology of the upper extremities in the control group, which comprised 44 upper extremities in 27 patients.

We found that patients with injuries of the C6-C7 and C5-C8 spinal segments had parameters of EMG amplitude for flexors of the hand and fingers that were approximately normal. Evaluation of the extensors’ status showed that EMG amplitude was reduced by 20%. Patients with injuries of the C5-Th1 spinal segments had decreased electrogenesis of hand and finger flexors (by 35%) and, to a greater extent, of finger extensors (by 47%) in 100% of the hands and fingers.

The amplitude of electrogenesis in the radial extensor of the wrist (m. extensor carpi radialis longus et brevis) was reduced in 100% of the wrists in all groups by a mean of 29% in patients with injuries at the C6-C7 spinal segmental levels, 35% in patients with injuries at C5-C8, and 49% in patients with injuries at C5-Th1. Normal values of electrogenesis amplitude for ulnar extensor of the wrist were observed in 52% of the wrists in the patients with injuries of C6-C7 and in 48% with injuries of the C5-C8 spinal segments, wherein the degree of reduction varied from 14% to 29%; in the group with injury of the C5-Th1 spinal segmental levels, it was reduced in 100% of the wrists on average by 43%.

All patients with wrist joint deformities in AMC had pathological patterns of electrogenesis, namely, hypersynchronous, partially decreased, slowing with a tendency to paling, and paling-like. In patients with injuries of the C6-C7 spinal segments, 45% exhibited hypersynchronous EMG, 34% exhibited partial decreases, and 21% exhibited paling-like EMG. In the patients with injuries of the C5-C8 spinal segments, most had decreased EMG, 56% had a tendency to paling, 23% had hypersynchronous EMG, and 22% had paling-like EMG. In children with injuries of the C5-Th1 spinal segments, 82% had decreased EMG with a tendency to paling (with rare frequency of oscillations), and 19% had hypersynchronous EMG (with rare frequency and high amplitude of oscillations).

Evaluation of the M-responses during stimulation of motor fibers of the upper extremities showed that in the patients with injuries of the C6-C7 spinal segments, 35% exhibited decreased amplitude of M-response. A decrease in M-response was observed in 29% of the patients along the radial nerve and in 37.5% of the patients along the ulnar nerve. The degree of amplitude reduction ranged from 20% to 60% relative to the reference parameters. The nerve conduction velocity (NCV) parameters in all patients were normal. In patients with injuries of the C5-C8 spinal segments, 51% had decreased amplitude of M-response. Decreased amplitude of M-response was noted in 62% of the patients along the radial nerve, in 76% of the patients along the ulnar nerve, and in 20% of the patients along the median nerve; the degree of amplitude reduction varied from 30% to 80% of normal values. The children in these groups had a slight decrease in NCV (on average by 23%).

In the group of patients with injuries of the C5-Th1 spinal segments, decreased amplitude of the M-responses was observed in 80% of the patients, which included 90% of the patients along the radial and ulnar nerves, and in 56% along the median nerve. The degree of amplitude reduction for M-responses ranged from 40% to 95%. A reduction of NCV for motor fibers was also noted in 25% to 43% of the age standards.

The degree of M-response decrease, EMG amplitude decrease, and structural impairment of the surface EMG in the forearm muscles was dependent on the number of involved segments but also reflected the degree of neuromuscular damage, which allowed prediction of the results of treatment.

Therapeutic approach

The aim of conservative and surgical treatment of wrist joint deformity was to fix the wrist in the functional position, to improve the gripping function, provide the possibility of self-care in patients, and improve the cosmetic appearance of the hand.

Non-surgical treatment

Non-surgical treatment of wrist joint deformation, which included application of a plaster or plastic splint, was made in the position giving the best possible contracture correction immediately following birth of the child. We recommended that the splint be worn for 20–22 hours a day during the first 3 months of life, removing them only for performing hygienic procedures, PT, and massage.

Surgical treatment

If non-surgical treatment was ineffective, the child underwent surgical treatment after they reached the age of six months.

In determining the sequence of upper extremity deformity treatment, the key point was to assess the passive movements in the ulnar joint. If a patient with extension contracture had ulnar joint passive flexion <90°, the first stage was to perform mobilizing surgery on the ulnar joint, which allowed better range of motion in the joint, followed by treatment of wrist joint contracture. In patients with passive flexion in the ulnar joint ≥90°, the first stage was to perform surgery on the hand to improve self-care capabilities. In patients with flexion contracture of the ulnar joint >90°, the first stage involved restoration of the extension in the joint prior to treatment of wrist joint deformation. In patients with pronational contracture of the shoulder (forearm) and in children over three years old, the first stage was to restore the limb position, followed by surgery on the hand. In the presence of concomitant deformation of fingers, their surgical treatment was combined with wrist joint surgery, which allowed restoration of the gripping function as soon as possible.

Tendon and muscle plasty of the forearm and wrist were performed in patients with passive correction of wrist joint contracture to the middle position or position of overcorrection in the sagittal plane and ≤15° of ulnar deviation in the frontal plane. In patients with passive correction of the wrist joint ≤10°–15° with palmar flexion and in the frontal plane of >15° ulnar deviation, tendon and muscle plasty were performed with corrective osteotomy of the carpal bones. In patients with wrist joint flexion contracture ≥90°and patients with severe flexion contracture of fingers in combination with pronounced shortening of the flexors of the hand and fingers, tendon and muscle plasty were performed with corrective osteotomy of the carpal bones and shortening osteotomy of the forearm bones.

In total, 90 patients with AMC aged 6 months to 18 years were treated (162 hands) for deformities of the wrist joints. Most patients underwent surgery between the ages of 1 to 6 years. Major surgery included tendon and muscle plasty of the forearm and hand (110 hands, 68%), tendon and muscle plasty with corrective osteotomy of the wrist bones (51 hands, 31%), and tendon and muscle plasty with corrective osteotomy of the wrist bones and shortening osteotomy of the forearm bones (1 hand, 1%). In the patients with injuries of the C6-C7 and C5-C8 spinal segments, the majority of patients received tendon and muscle plasty of the forearm and hand. In the group with C5-Th1 injuries, the frequencies of tendon and muscle plasty alone and in combination with corrective osteotomy of the wrist bones were similar, and one patient underwent tendon and muscle plasty with corrective osteotomy of the carpal bones and shortening osteotomy of the forearm bones (Table 1).

In all patients who underwent tendon and muscle plasty of the forearm and hand, the most prevalent age was younger than 3 years old. Patients older than 3 years with injuries of the C6-C7 and C5-C8 spinal segments underwent tendon and muscle plasty at a higher rate. In 68% of the children of the same age with injuries of the C5-Th1 spinal segments, surgery included corrective osteotomy of the carpal bones.

Table 1. Options for surgical interventions in patients with different levels of spinal segmental injury

Options for surgical interventions

Level of spinal segmental lesion

Total

(%)

С6-С7

С5-С8

С5-Th1

Tendon and muscle plasty of the forearm and hand

25 (76 %)

51 (82 %)

34 (51 %)

110 (68 %)

Tendon and muscle plasty with corrective osteotomy of the wrist bones

8 (24 %)

11 (18 %)

32 (48 %)

51 (31 %)

Tendon and muscle plasty with corrective osteotomy of the wrist bones and shortening osteotomy of the forearm bones

0

0

1 (1 %)

1 (1 %)

Total

33 (100 %)

62 (100 %)

67 (100 %)

162 (100 %)

Additional surgeries to treat associated upper limb deformities combined with surgeries on the wrist joint include: treatment of adduction–flexion contracture of the first finger, flexion contractures and ulnar deviation of the triphalangeal fingers, and de-rotational osteotomy of the radial bone.

During the surgeries, we found that the degree of degenerative changes in the tendons of the hand flexors and extensors as well as the unity of the tendons to the underlying tissues depended on the number of involved spinal segments. Thus, in patients with contractures of the wrist joints with injuries of the C6-C7 and C5-C8 spinal segments, it was possible to recover active extension of the hand, but in patients with C5-Th1 injuries, only the removal of the hand in a functionally beneficial position improved the gripping function of the hand and restored a minimum ability of self-care.

Immobilization with a plaster splint and fixation of the hand with wires after tendon and muscle plasty on the forearm and hand were performed within 4 weeks, regardless of the child’s age. In patients with tendon and muscle plasty and a corrective osteotomy of the wrist bones with immobilization of 5–6 weeks, the wires were removed after x-ray radiography indicated control without a plaster splint and if there were signs of consolidation in the area of the osteotomy. In those cases, immobilization was an average of 6–8 weeks after tendon and muscle plasty of the forearm and wrist in combination with corrective osteotomy of the wrist and shortening osteotomy of the forearm bones.

After limb immobilization, rehabilitative treatment was prescribed, including PT (thermal procedures, electrophoresis or phonophoresis with lydasum in the region of postoperative scarring, electrical stimulation of the hand and finger extensors, and flexor digitorum), and physical therapy and massage were also provided. To develop passive movements in the wrist joint and finger joints, we used devices for mechanical therapy (ARTROMOT®-F; ORMED, Germany and THERA Trainer®; Medica, Germany) and a rehabilitation complex with enhanced biological feedback (Armeo® Spring Pediatric; Hocoma, Switzerland), which helped to speed up the process of forming new movement patterns and improve the possibility that the patient could perform self-care. After cessation of plaster immobilization and removal of the wires from the hand, a removable joint-immobilizer from the middle third of the forearm to the metacarpophalangeal joints was made, with fixation of the wrist joint in the position of the dorsal extensions of 10°–15° to be worn constantly for 6 months.

Results

Results of surgical treatment were obtained for 90 patients (162 hands) with AMC and deformities of the wrist joint and different levels of spinal segmental injury who were followed-up with treatment from 6 months up to 8 years. The evaluation of results was performed by taking into account the following criteria: the hand position in the frontal and sagittal planes, amplitude of active and passive movements in the wrist joint, cosmetic appearance, and functional ability of the hand to grip. The functional position of the hand in the sagittal plane was considered to range from 0° to 30° of dorsal extension in the sagittal plane and from 0° to 15° of ulnar deviation in the frontal plane. According to the literature, an amplitude of 35° is sufficient for performing everyday routine hand functions (from 5° flexion to 30° extension in the sagittal plane) [18]. After surgical treatment, the hand position in all cases of wrist joint deformity in patients with various spinal segmental level injuries improved an average of 51°– 4° of palmar flexion (in the sagittal plane) and of 33°–5° of ulnar deviation (in the frontal plane) (Table 2).

Along with the change in hand position, the amplitude and sectors of passive and active movements in the wrist joint also changed (Table 3).

The amplitude of passive movements in the wrist joint after surgical treatment for flexion contractures of the wrist joint and isolated ulnar deviations of hands decreased by 37% and 43%, respectively, with elimination of flexion contractures in the wrist combined with ulnar deviations of hands by 18%. The amplitude of active movements after surgery decreased by 28% in flexion contracture of the wrist joint and flexion contracture of the wrist joint combined with ulnar hand deviation, and by 44% in ulnar deviation of hands. Despite the decrease in the amplitude of passive and active movements in the wrist joint, sector movements after surgery improved into the functional range in all types of wrist joint deformities, which improved the ability of the hand to grip and the chances of self-care in the patients.

Evaluation of the functional ability of the hand to grip showed improvements in patients with different levels of spinal segmental injury (Table 4). The functional ability of the hand to grip improved by 19% in the C5-C8 spinal segmental injuries and by 21% in the C5-Th1 segmental injuries after surgical treatment, due to improvement in all investigated types of gripping, and was classified as high and moderate functional ability of the hand to grip, respectively. In the patients with C6-C7 spinal segmental levels after surgical treatment, only an improvement of 3% was observed, mainly because the morphogenetic gripping cosmetic appearance improved in all patients.

To perform an objective assessment of the hand state after surgical treatment, we developed a scale that included analysis of the following characteristics: the hand functional position in the frontal and sagittal planes, amplitude of active hand extension, cosmetic appearance, and functional ability of the hand to grip in points. Treatment outcome was regarded as good in 53% of the hands, satisfactory in 42%, and poor in 5%, by total points. Analysis of treatment outcomes depending on the injury of the spinal segmental level showed that 100% of the patients with a C6-C7 level injury after surgical treatment had good treatment outcomes and that 84% and 16% of the patients with C5-C8 injuries had good and satisfactory outcomes, respectively. In addition, 11% of the patients with injuries of the C5-Th1 spinal segmental level had good treatment outcomes after treatment in, 79% had satisfactory outcomes, and 10% had unsatisfactory outcomes.

After treatment, 12% (19 hands) showed recurrence of deformation within an average of 2.5 years after surgical intervention, and 1% (2 hands) showed formation of secondary deformations. Analysis showed that 3% (1 hand) of the patients with C6-C7 spinal segment injuries, 9% (6 hands) of the patients with C5-C8 injuries, and 18% (12 hands) of patients with C5-Th1 injuries had recurrences of deformities. Secondary deformation in the form of extension contracture of the wrist joint (2 hands) was observed in 1 patient with a lesion of C6-C7 spinal segmental level.

Conclusions

The developed therapeutic approach for wrist joint deformities in children with AMC improved the gripping function and cosmetic appearance of the hand as well as the self-care capabilities of the patients. Determining the level of injury on the cervical intumescence in this group of patients enabled prediction of the treatment outcome for wrist joint deformity. In the patients with injuries of the C6-C7 and C5-C8 segments, it was possible to restore active extension of the hand, but in patients with injuries of the C5-Th1 spinal segments, it was only possible to remove the hand in a functionally beneficial position. With the increasing number of involved spinal segments in various pathological processes, the functional state of the hand will show greater deterioration, and the number of unsatisfactory outcomes and postoperative recurrences after surgical treatment will increase.

Evgeniya A Kochenova

The Turner Scientific and Research Institute for Children’s Orthopedics

Author for correspondence.
Email: fake@eco-vector.com

Russian Federation MD, orthopedic and trauma surgeon. 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

Margarita V Savina

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: fake@eco-vector.com

Russian Federation MD, PhD, research associate of the laboratory of physiological and biomechanical research. The Turner Scientific and Research Institute for Children’s Orthopedics

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