Congenital radioulnar synostosis: symptom complex and surgical treatment

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Abstract


Background. Congenital radioulnar synostosis (CRUS) is a rare musculoskeletal disease with a wide-ranging symptom complex. Attitudes toward surgical treatment of the disease is very diverse, ranging from complete negation to acceptance. When choosing a treatment method, high recurrence and complication rates should be taken into account.

Aims. To analyze the clinical implications of CRUS and to identify optimal treatment options.

Materials and methods. From 2008 to 2015, 54 patients (31 boys and 23 girls; aged 1–14 years) with CRUS were examined and treated. Presenting complaints and the possible factors leading to disease development were investigated; orthopedic examination, roentgenography, electromyography, and computed tomography were performed. The treatment approach was determined on the basis of the clinicoroentgenological presentation.

Results. All cases of CRUS were sporadic. In 43.7% patients, risk factors resulting in disease development were detected. Unilateral lesions were observed in 30 patients, whereas bilateral lesions were observed in 24 patients. According to the Cleary and Omer classification, the first type is the rarest; it is distinguished by the absence of bony fusion and close to average forearm positioning. In such cases, operative treatment is not necessary. For the second and third types, pronounced pronation forearm realignment requiring corrective derotational osteotomy of the radial bone is the main factor. For the fourth type, the main functional disorder is the restriction of the forearm flexion; treatment for this type involves resection of the radius head. We attempted to divide the synostosis 
(to achieve active movements) in five patients; however, we were unsuccessful. In three patients, synostosis recurrence occurred; and in two patients, active movements were not obtained after surgery. In four patients, radial nerve neuropathy was detected in the postoperative period after conservative therapy. In two patients, ulnar fractures occurred as a result of a fall; in one of these patients, fragment apposition was required.

Conclusions. Clinicoroentgenological manifestations of CRUS determine the treatment options. The most typical and important of these manifestations is the pronation positioning of the forearm. In such cases, it is reasonable to start operative CRUS treatment after 3 years. All variants of deformation are indicators for operation, and treatment options are determined by the degree of severity of the deformation. Attempts to form the forearm bone neoarthrosis in order to get rotational movements is not effective and can result in deformation recurrence.


Background. Congenital radioulnar synostosis (CRUS) is a rare musculoskeletal disease with a wide-ranging symptom complex. Attitudes toward surgical treatment of the disease is very diverse, ranging from complete negation to acceptance. When choosing a treatment method, high recurrence and complication rates should be taken into account.

Aims. To analyze the clinical implications of CRUS and to identify optimal treatment options.

Materials and methods. From 2008 to 2015, 54 patients (31 boys and 23 girls; aged 1–14 years) with CRUS were examined and treated. Presenting complaints and the possible factors leading to disease development were investigated; orthopedic examination, roentgenography, electromyography, and computed tomography were performed. The treatment approach was determined on the basis of the clinicoroentgenological presentation.

Results. All cases of CRUS were sporadic. In 43.7% patients, risk factors resulting in disease development were detected. Unilateral lesions were observed in 30 patients, whereas bilateral lesions were observed in 24 patients. According to the Cleary and Omer classification, the first type is the rarest; it is distinguished by the absence of bony fusion and close to average forearm positioning. In such cases, operative treatment is not necessary. For the second and third types, pronounced pronation forearm realignment requiring corrective derotational osteotomy of the radial bone is the main factor. For the fourth type, the main functional disorder is the restriction of the forearm flexion; treatment for this type involves resection of the radius head. We attempted to divide the synostosis 
(to achieve active movements) in five patients; however, we were unsuccessful. In three patients, synostosis recurrence occurred; and in two patients, active movements were not obtained after surgery. In four patients, radial nerve neuropathy was detected in the postoperative period after conservative therapy. In two patients, ulnar fractures occurred as a result of a fall; in one of these patients, fragment apposition was required.

Conclusions. Clinicoroentgenological manifestations of CRUS determine the treatment options. The most typical and important of these manifestations is the pronation positioning of the forearm. In such cases, it is reasonable to start operative CRUS treatment after 3 years. All variants of deformation are indicators for operation, and treatment options are determined by the degree of severity of the deformation. Attempts to form the forearm bone neoarthrosis in order to get rotational movements is not effective and can result in deformation recurrence.

Keywords: radioulnar synostosis, upper limb congenital anomalies, surgical treatment.

Introduction

Congenital radioulnar synostosis (CRUS) is a rare musculoskeletal disease that was first described by Sandifort in 1793. The frequency of CRUS among all congenital upper limb deformities ranges between 1.12% [1] and 9.35% [2]. Among other musculoskeletal deformities, the frequency of CRUS ranges between 0.11% [3] and 0.61% [2]. Usually, cases of CRUS are sporadic, but in a number of instances, inherited synostosis cases have been reported. For example, Kolyaditski (1967) described a family in which CRUS was present in four generations accounting for a total of 14 relatives. Of the 14 relatives, 13 were men [4]. Abalmasova and Luzina (1976) noted CRUS cases in two families out of six observations [5]. A number of authors have reported a larger occurrence of the disease in boys [2, 6]. However, some reports have also indicated that the disease occurrence does not depend on sex [7].

Frequently, CRUS is combined with other orthopedic and somatic pathologies. Among the concomitant orthopedic pathologies, the most frequently observed is a lesion of the opposite limb in the form of synostosis or rotational forearm contracture. According to the literature, different ratios between bilateral and unilateral synostosis have been noted: Chaklina reported a 4:1 ratio [8]; Simmons and Southmayd (1983), 3:2 [7]; and Green and Mital (1979), 2:1 [9]. Other orthopedic pathologies that can present in combination with CRUS are scoliosis, syndactyly, hypoplasia of the thumb, congenital dislocated hip, knee joint anomalies, clubfoot, polydactyly, carpal bone concrescence, and joint hypermobility. Some of the related syndromes are Apert, Carpenter, Williams, Klinefelter, Holt Oram, acrocephalosyndactyly, and arthrogryposis syndromes. Mandibular facial dysostosis, microcephaly, multiple exostosis, as well as cardiac, renal, and neurological anomalies have been noted [7].

The Cleary and Omer classification [6, 7, 10, 11] for CRUS is the most widely used and includes the following types: fibrous synostosis, bone synostosis with the radial head hypoplasia, bone synostosis with posterior dislocation of the radial head, and bone synostosis with anterior dislocation of the radial head.

Currently, the attitude towards surgical intervention is controversial among orthopedic surgeons. Some authors believe that in most cases of synostosis, there is no need for surgical treatment [6]. Green (1979) recommended postponing the decision of undergoing surgery until the patient has a notion regarding his/her future profession [9]. However, Ogino and Hikino (1987) examined 40 CRUS patients and showed that more than 60% of patients with deformities presented complaints on discomfort in everyday life. Among their patients, none complained of pronation position up to 20° [12].

The recommended age for surgical treatment among patients with CRUS ranges from 2 years [13] up to adulthood [14]. The goals of the treatment are to achieve movement of the forearm out of a pronation position, creation of mobility between the radial and ulnar bones, and increasing the movement amplitude in the elbow joint. More than 20 single-step and multistage methods and their modifications are known today, and at least 16 types of interposed materials have been described [2]. The most popular method is the elimination of the pronation position of the forearm by osteotomy of the (radial) bone distal to the synostosis [1], including lining the separation zone with heterofascia [3], autohomofascia or a plastic liner [15], with interposition of muscle and fascia [16]. A silicone cap is placed on the radial osteotomy surface [17]. The radial bone osteotomy at two levels has been proposed in cases of shortage of a derotational effect [13]. Double-level bone osteotomy a [6, 18, 19] and derotational osteotomy at the synostosis level are also used [7, 9, 12, 20].

The Ilizarov’s apparatus is used in large (more than 90°) pronation contractures [10] and in cases of neoarthrosis development between the forearm bones [13, 21, 22]. In a number of cases, when the rotator functions are preserved, a longitudinal dissection is performed at the bone block area between the radial and ulnar bones. Then, the area is closed by demineralized osteochondral allotransplants, and adjoining soft tissues are inserted between the bones [23].

If patients complain of pain, clicks, or movement restriction in the elbow joint, in cases of anterior dislocation of the radial head (fourth type of synostosis by Cleary and Omer classification), the dissection of the lateral capsule of the elbow joint [24] and resection of the radial head are performed [11].

Many authors have reported a high frequency of neurological complications related to osteotomy and forearm bone rotation [15]. These are paralysis of the radial and median nerve, adhesion, and recurrence [2]. The resection of a skin part in the osteotomy zone contributes to fewer complications during the postsurgical period [12]. Mobilization of soft tissues makes it possible to avoid or decrease the risk of complications [12, 25]. In case of complications, dissection of the bandage is proposed. If this is not sufficient, then wire removal is recommended [7, 26]. A larger derotation is associated with a greater frequency of complications. For example, for instrumental correction, when attempts were made to eliminate a pronation position of more than 90°, the frequency of complications increased from 25% [21] to 50% [10]. The attempt to line the osteotomy zone with a fascial-fatty flap usually fails, leading to the restoration of a bone bridge between the forearm bones [7]. Attenuation of rotational movements occurs within 6–12 months [16].

Materials and methods

Between 2008 and 2015, we treated 54 CRUS patients within the ages of 1–14 years at The Turner Scientific and Research Institute for Childrens Orthopedics. All patients signed the informed consent for participation in the study and surgical intervention.

The complaints and factors that could lead to disease development were investigated. During the orthopedic examination, we measured the segment lengths and amplitude of movements in the upper limb joints. Patients were assessed to determine the presence of concomitant orthopedic and somatic pathologies. The roentgenograms were studied closely for synostosis localization, length, limb bone deformity, adjacent joint condition, and bone age. In a number of cases, electromyography and computed tomography were also performed.

All patients had the bone type fusion according to Cleary and Omer classification [27]. All patients enrolled in this study underwent surgical treatment. The surgical outcomes were assessed in a period from 6 months to 10 years. Two patients with bilateral CRUS had a fibrous type fusion on one side in a functionally advantageous position.

Results

A total of 54 CRUS patients were treated at our institute. All cases were sporadic. Risk factors for CRUS development were detected in 43.7% of patients. These were pregnancy-related factors (toxicosis, especially in the first trimester of pregnancy, and placental abruption) and chronic gynecologic and extragenital pathologies.

Patients were 31 boys and 23 girls, which corresponds with the observations of other researchers. Unilateral lesion was noted in 30 patients and bilateral lesion in 24 patients. Frequently, in 36.7% of the cases (11 children) of unilateral CRUS, supination–pronation contracture of the opposite forearm was present, but there was no radiographic confirmation of bone disorders.

Three patients underwent surgical treatment before admission to the Turner Institute. One of them underwent derotational osteotomy of the radial bone in the middle third with fixation with plate and screws. A 30° pronation position of the forearm was obtained. The second patient underwent dissection of the proximal radial bone. As a result, in three years, passive rotational movements were obtained: 5° supination, 3° pronation; no active movements were achieved. The two patients had bilateral CRUS and were admitted for the elimination of untreated limb deformity. The third patient underwent the segmental resection of the proximal radial bone, derotational osteotomy and intramedullary osteosynthesis of the ulnar bone, with mounting of the external fixation device on the left forearm with subsequent distraction. Two years after admission, the patient complained of discomfort in the wrist joint. Clinically, the patient developed radial club hand, which was caused by sagging of the radial bone.

The absence of the forearm rotation is the main manifestation of the disease. The degree of the functional insufficiency depends on the forearm pronation position angle. The rotational hypermobility of the wrist joint partly compensates the lack of the forearm rotation. The vast majority of children with a unilateral lesion presented forearm shortening by 1–2 cm, upper arm shortening by 1 cm (two patients), and wrist shortening by 1 cm (one patient). A significant valgus deformity (more than 20°) was noted in four elbow joints; a significant varus deformity (5° and 1°) was noted in two elbow joints.

Among other orthopedic pathologies, planovalgus foot deformity, hip dysplasia, and chest and spinal column deformity were detected.

The allocation of patients according to the radiographic types is shown in Table 1.

Table 1

Value of the forearm pronation position depending on the congenital radioulnar synostosis type

Pronation angle (°)

Type 1

Type 2

Type 3

Type 4

<3

100%

16.67%

6.9%

75.%

3–59

0

8.33%

10.33%

12.5%

6–89

0

41.67%

55.17%

12.5%

>9

0

33.33%

27.6%

0

Total

100%

100%

100%

100%

The clinical presentation of type 1 radioulnar synostosis (syndesmosis) did not include pronation contracture, or it was insignificant. There were no forearm rotational movements, or they were of the swinging type. In one case, the radial head was unchanged and decentered towards the humerus condyle head. In another case, the radial head had a small skewness forward and laterally, and it was projected onto the condyle head. The metaphysis of the radial bone was thickened. The proximal radioulnar joint was narrowed in both patients and the articular surface was eroded. Other osseous structures remained unchanged (Fig. 1). In this type of synostosis, surgical treatment was not indicated.

In type 2 synostosis, the proximal forearm bone branch was represented by a common conglomerate. The radial head was not differentiated (Fig. 2).

Type 3 was characterized by posterior or posteroexternal dislocation of the radius. The radial head was hypoplastic. Its breadth narrowed and there were no anatomical bends of the neck. The growth zone was not defined or was displaced (Fig. 3).

In types 2 and 3 synostosis, the radial shaft had an arcuate shape with an angle open towards the ulnar side. These were the most severe types as the forearm was at the maximal pronation position. It exceeded 60° in 75.0% and 82.8%, respectively. In the clinical presentation, 36.6% of cases had moderate (by 10–30°) elbow extension restriction.

In type 4 synostosis, the enlargement of the radial bone metaphysis (fusion area with the ulnar bone) was characteristic. At that level, the radial bone sharply shifted forward. Thus, the head was dislocated anteriorly (Fig. 4). This deformity caused elbow flexion restriction in 33.3% of cases.

The radial shaft arc in CRUS in our patients was enhanced. While the physiological bend in the frontal plane is 13° on average [28], in 27.7% of children with CRUS it is 20° or more. In 22.2% of the cases, the bend exceeds 10° (on average 4°) in the sagittal plane [28]. In three cases, the radius diameter was twofold the diameter of the ulnar bone.

The wrist joint in CRUS had three levels of radial and ulnar bone location: 68.6% present the (0) variant (articular plates located at the same level), 5.7% present the (–) variant (the ulnar head was located proximal to the radius articular plate); and 25.7% present the (+) variant (the ulnar bone protrudes distally of the radius), which generally corresponds to frequency variation in healthy persons [29].

Six patients (11.1%) presented dorsal subluxation of the ulnar bone. In 7.4% of the CRUS limbs, the bone age retardation was approximately 1–2 years. The electromyographic study showed that in the forearm round pronator muscle on the lesion side, the electroactivity was 232.6 ± 12.4 µV. On the unaffected side, it was 345.3 ± 9.5 µV (р < 0.05). In the supinator muscle, it was 208.3 ± 14.1 µV and 344.4 ± 15.6 µV (р < 0.05) on the lesion side and unaffected side, respectively. The potential frequency on the side of the lesion was decreased on average by 5% compared with the unaffected side. “At rest” electroactivity was not detected in either of the children. Thus, on the lesion side, the contractile function of the studied muscles was decreased compared with the unaffected side, which may be caused by the lack of the forearm rotator functional load.

The types of the surgical intervention indicated are shown in Table 2.

Table 2

Surgical intervention types by different types of congenital radioulnar synostosis

Surgery name

Type 2

Type 3

Type 4

Total

Derotational osteotomy of the radius (including two-level)

14  

30

1

45

Synostosis elimination

1

 

0

4

5

Th e radial head resection

0   

 

0

3

3

Others*

3

0

0

3

Total

18

 

30

8

56

*Note: Corrective supracondylar osteotomy of the humerus, correction of secondary forearm deformation using distraction device.

The main goal of the surgical treatment was the elimination of the functionally unfavorable pronation position of the forearm. A pronation position of the forearm greater than 60° was considered to be the indication for surgery.

After performing an incision in the upper third of the forearm, derotational osteotomy of the radius distal of the synostosis was performed. The interosseous membrane along the radial shaft was dissected. The forearm was set in the pronation position at 0–10°, and the position was locked by transversely drawn Kirshner wires or biodegradable screws (Fig. 5). If the correction failed, an incision in the lower third of the forearm was made, and the radius was transected, which allowed us to increase the rotation degree. In this case, the radius fragments were also locked by an intraosseous wire (Fig. 6). After derotational osteotomy of the radius, the position was locked with plate and screws for social reasons in two cases (one patient). In the postsurgical period the forearm was immobilized using a splint until bony callus formation.

In five cases, interventions were performed with the main goal of not only eliminating the contracture, but also restoring the rotational movements. After synostosis elimination, a radioulnar joint was formed. In one case, this was achieved by using an annular band made of lavsan tape. In three cases, dura matter was used for lining the neoarthrosis zone. In another case, the zone was lined with local fasciocutaneous tissues. Although good rotational movement amplitude was obtained on the operation table, in the immediate postsurgical period, the active amplitude was 20°–25°, and the passive rotational movement amplitude was up to 15°. In 1–1.5 years postoperatively, all patients presented a decrease of the active movement amplitude. Two patents retained rotational movement of up to 30°. In the rest of the cases, synostosis formed in a functionally favorable position.

Three patients with anterior dislocation underwent radial head resection, which allowed an increase in elbow flexion. One girl with valgus deformity and elbow joint instability combined with CRUS underwent corrective supracondylar osteotomy of the humerus.

Children who had pain syndrome and radial club hand that resided in the vicinity of the institute underwent a two-stage method to eliminate the faulty position. First, the radius was directed downward using a distraction device to create correct relations in the wrist joint. Then, upon removal of the device, the proximal radius part was matched and locked to the ulnar bone.

The surgical treatment outcomes were assessed from 6 months to 10 years postoperatively. A result was considered optimal if after a derotational surgery, the forearm acquired a pronation position from 0 to 20° or if after resection of the radial head, the movement amplitude of the elbow joint increased by 20° or more. A result was considered to be satisfactory if complications developed and led to prolongation of the treatment period but did not affect the surgical outcome. A result was considered to be unsatisfactory when a patient’s functional capabilities remained unchanged or worsened after treatment.

A total of 45 radius osteotomies were performed. In all patients, a long-term functionally advantageous pronation position (less than 20°) was achieved. Four patients (8.9%) developed a transient neuropathy of the radial nerve in the postoperative period after derotation by 70°–90°. The neuropathy was resolved with drug therapy. Two patients (4.4%) with a plaster cast fell and fractured the ulnar bone. In one case, the displacement did not affect the final outcome. In the other case, a repositioning with additional wire fixation was required. Eventually, the goal was achieved; the bones fused in apposition of rotational correction without angular deformity.

The attempt to create a neoarthrosis between the radius and ulnar bone failed; that is, active rotational movements were not obtained. However, the forearm was repositioned medially. That is, the surgery can be designated as a variant of derotational osteotomy of the radius. Thus, after derotational osteotomy of the radius and creation of neoarthrosis between the forearm bones, 88.0% of the outcomes were optimal, and 12.0% were satisfactory. After resection of the radial head, the movement amplitude increased by 20° or more that was equivalent to an optimal result in all operated patients. There were no unsatisfactory results.

Discussion

It can be assumed that CRUS is present based on the pronation position degree. When the deformity is pronounced, parents and pediatricians can see the obvious and unusual position of the forearm as early as in the maternity clinic. However, it is also common that parents and children notice the abnormalities later on, particularly when the child begins to use their hands. This deformity can translate into difficulty in carrying out self-maintenance tasks such as holding water in palms, incorrectly holding a spoon, difficulty combing hair, buttoning up the shirt, among others. In one case, the physical education teacher was the first to notice that a schoolboy was unable to perform a task. At an older age, children tend to complain about pain associated with loading. Thus, pronounced pronation position of the forearm causing self-maintenance failure is the most frequent CRUS manifestation.

With a greater deviation, indication for surgical treatment becomes clearer. Regarding the need of surgical correction, we do not see the point in waiting until a man or women decides on a future profession. Instead, we advocate that early treatment is ideal for more active use of the hand as well as for physical and mental growth of the child. At the same time, one must consider the frequency of neurological complications that can occur as a consequence of derotation. In the postsurgical period, it is necessary to assess the sensitivity and active movement of the fingers in a splint. Patient that are too young may sometimes be incapable of performing important movements required by the physician, which may be useful for detecting a sensitivity disorder; therefore, we consider it appropriate to start surgical treatment after 3–4 years of age.

Based on our experience and that of our foreign colleagues, the success rates for neoarthrosis creation between the forearm bones are low. As a result, in 1 or 1.5 years, the achieved movement amplitude is decreased and synostosis recurs. This applies particularly to cases of synostosis types 2 and 3 according to the Cleary an Omer classification.

The main goal of surgical treatment is achieving a forearm pronation position of 0–20°. The remaining rotational deficit is compensated by movements in the shoulder joints and hypermobility of the wrist joint typical in such patients. Forced rotation at one level can lead to the nerve traumatization. If derotational osteotomy of the radius and dissection of the interosseous membrane do not lead to the desired outcome, then osteotomy in the lower third of the radius may be necessary for neuropathy prevention. Rotation at one level, including the use of distraction-rotational devices, has complications of up to 50% [10]. According to our observations, a derotation of 70–90° can result in a transient neuropathy of the radial nerve in 8.9% of the cases.

Conclusions

1. CRUS is a sporadic disease attributed to exogenic and endogenic factors (maternal conditions during pregnancy).

2. The indication for surgical treatment is based on the degree of the pronation position of the forearm, the deviation angle at the level of the elbow joint, and the elbow joint function restriction.

3. The clinical manifestations of the pathology are associated with the type of forearm bone fusion and the radial head position.

4. It is reasonable to start CRUS surgical treatment at the age of 3 years.

5. All deformity variants are indications for surgical treatment, considering that treatment technique selection is determined by the degree of its severity.

6. The attempts to create neoarthrosis of the forearm bones in order to obtain rotational movements are inefficient and dangerous because of possible deformity recurrence.

Information on funding and conflict of interest

This work was supported by The Turner Scientific and Research Institute for Childrens Orthopedics, Saint Petersburg, Russian Federation. The authors declare no conflict of interests.

Evgeny V Prokopovich

The Turner Scientific and Research Institute for Children’s Orthopedics

Author for correspondence.
Email: afonichev@list.ru
MD, PhD, orthopedic surgeon of the department of trauma sequelae and rheumatoid arthritis. Th e Turner Scientifi c and Research Institute for Children’s Orthopedics.

Mikhail A Konev

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: afonichev@list.ru
MD, chief of the department of trauma sequelae and rheumatoid arthritis. The Turner Scientifi c and Research Institute for Children’s Orthopedics

Konstantin A Afonichev

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: afonichev@list.ru
MD, PhD, professor, head of the department of trauma eff ects and rheumatoid arthritis. The Turner Scientifi c and Research Institute for Children’s Orthopedics, Saint-Petersburg, Russian Federation.

Ivan E Prokopovich

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: afonichev@list.ru
MD, resident of the Turner Scientifi c and Research Institute for Children’s Orthopedics

Aleksander B Kovzikov

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: afonichev@list.ru
MD, orthopedic and trauma surgeon of the department of trauma eff ects and rheumatoid arthritis. Th e Turner Scientifi c and Research institute for Children’s Orthopedics.

Maksim S Nikitin

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: afonichev@list.ru
MD, orthopedic and trauma surgeon of the department of trauma effects and rheumatoid arthritis. The Turner Scientifi c and Research Institute for Children’s Orthopedics.

Vladimir V Selizov

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: afonichev@list.ru
MD, orthopedic and trauma surgeon of the department of trauma eff ects and rheumatoid arthritis . The Turner Scientifi c and Research institute for Children’s Orthopedics.

Tatyana S Vinokurova

The Turner Scientific and Research Institute for Children’s Orthopedics

Email: afonichev@list.ru
MD, PhD, leading research associate of the laboratory of physiological and biomechanical research. The Turner Scientifi c and Research Institute for Children’s Orthopedics

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