Reconstruction of a postraumatic radial club hand

Received: 8 March 2016

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Revised: 16 June 2016

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Accepted: 8 July 2016

DOI 10.1002/micr.30087

CASE REPORT

Reconstruction of a postraumatic radial club hand with a free fibular osteoseptocutaneous flap and Sauve–Kapandji procedure—A case report Ricardo Horta1,2 | Ricardo Nascimento1,2 | Alvaro Silva1,2 | Rui Pinto3 | Pedro Negr~ao3 | Ricardo S~ao-Sim~ao3 | Jorge Carvalho1,2 | Marta Santos Silva1,2 | Jose Amarante1,2 1 Department of Plastic, Reconstructive and Maxillo-Facial Surgery, and Burn Unity, Centro Hospitalar De S~ao Jo~ao, Faculty of Medicine, University of Porto, 4202-451 Porto, Portugal

Alameda Professor Hern^ani Monteiro, Porto, Portugal

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Orthopedic Department, Centro Hospitalar S~ao Jo~ao, Faculty of Medicine, University of Porto, Porto, Portugal Correspondence res, n 234, Ricardo Horta, Avenida Mene Bloco 2, 4 Frente Esquerdo, 4450-189, Matosinhos Sul- Porto, Portugal. Email: [email protected]

Abstract Radial club hand may be congenital or acquired; radial deviation of the hand is usually found, associated with palmar flexion–pronation and treatment of severe forms of radial club hand is often difficult. Here we present a case of reconstruction of a severe postraumatic radial club hand with a free fibular osteoseptocutaneous flap and Sauve–Kapandji procedure in a 28-year-old man. The patient had a radial deviation of the wrist and right upper limb shortening as a result of an infected pseudarthrosis of the radius. This deformity was reconstructed with a free fibular osteoseptocutaneous flap associated to arthrodesis of the distal radioulnar joint and an ulnar resection osteotomy –Kapandji procedure). proximal to the arthrodesis in order to restore rotation of the forearm (Sauve The flap fully survived and no complications were seen in the early postoperative period at both recipient and donor sites. Radius alignment was restored. At 5-month follow-up, the skeleton was healed. There was minimal osteopenia at the distal radial segment. Wrist extension was 48 degrees, flexion 24 degrees, and pronation–supination was 58–0–48 degrees, with full finger flexion. The patient could hold a 4 kg dumbbell with the elbow flexed without discomfort. His DASH score—Disabilities of the Arm, Shoulder, and Hand Questionnaire was 15.83. Combined free fibular osteoseptocutaneous flap and Sauve–Kapandji procedure may be considered in severe forms of postraumatic radial club hand, however, further data are necessary. KEYWORDS

procedure, radial club hand, reconstruction, Sauve–Kapandji, vascularized free fibula

1 | INTRODUCTION

Fitoussi, 2015). Several treatments to obtain lasting alignment of the hand relatively to the forearm have been described, such as radius

Radial club hand may be congenital or acquired. Congenital club hand

replacement, radialization to facilitate positioning of the carpus facing

affects the lateral part of the upper limb, with radial aplasia or hypopla-

the distal ulna, or centralization (de Jong et al., 2012; Romana et al.,

sia. Radial deviation of the hand is usually found, associated with pal-

2015).

mar flexion–pronation. This deformity is usually treated surgically in

A forearm fracture may be complicated by the disruption of the

infants and young children but the management of this problem in

distal radioulnar, proximal radioulnar, or radiocapitellar joints. Recon-

adults and in postraumatic situations is complex and challenging. This

struction of the distal radioulnar joint (DRUJ) is important to reduce

pathology may also occur after a forearm fracture, hematogeneous

the functional sequelae, as important capsuloligamentous structures

osteomyelitis of the radius (absorption and lysis can produce a radial

contribute to its overall stability, especially during transverse loading

defect) or after severe arthritis. It may also be secondary to surgical

with resisted elbow flexion (Cavadas & Thione, 2014). The guiding

procedures on the forearm (osteotomies among others) (de Jong,

principles for treating acute forearm fractures with associated DRUJ

Moran, & Vilkki, 2012; Patel & Paksima, 2010; Romana, Ciais, &

disruption are restoration and maintenance of anatomic alignment of

Microsurgery 2016; 00: 00-00

wileyonlinelibrary.com/journal/micr

C 2016 Wiley Periodicals, Inc. V

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Posttraumatic right radial club hand: (A) flexion; (B) extension; (C) anteroposterior; (D) lateral view: X-ray of the subacute injury showing pseudarthrosis of the radial diaphysis and radial deviation of the wrist. E: CT angiography showing the course of radial and ulnar artery along the forearm to the wrist

FIGURE 1

the bones, usually with open reduction and internal fixation, repair of

The patient was further referred to our department due to the

the DRUJ if necessary (unstable after fixation), and repair of associated

presence of a radial deviation of the wrist and right upper limb shorten-

soft tissue damage (George & Lawton, 2015).

ing which prevented the patient’s daily living activities (Figure 1). A

A case of reconstruction of a postraumatic radial club hand with a

reconstructive procedure using vascularized bone was then planned

–Kapandji free fibular osteoseptocutaneous flap associated to a Sauve

(Figure 2). First, the area of pseudarthrosis in the radial diaphysis was

(S-K) procedure is presented.

identified. A partial radial diaphyseal ostectomy (6 cm) was performed and an arthrodesis of the DRUJ was accomplished with two screws

2 | CASE REPORT

together with an ulnar resection osteotomy proximal to the arthrodesis in order to restore rotation of the forearm (S-K procedure). Next,

A 28-year-old man with no past medical history was involved in a

osteosynthesis of the radius was performed with a blocking plate and

bicycle accident during which he suffered a type I open fracture (Gus-

six blocking screws. The radius diaphyseal defect was reconstructed

tilo and Anderson classification) of the right forearm. He was initially

with a free fibular osteoseptocutaneous flap with a dorsal skin monitor-

treated with plate osteosynthesis of diaphyseal fractures of the distal

ing island, also important because of scar tissue release (Figure 3). The

radius and ulna. In the postoperative period, the patient developed an

peroneal artery was anastomosed end-to-end to the radial artery and

infection at the fracture site with skin necrosis and a split-thickness

the concomitant peroneal vein to the intermediate vein of the forearm.

skin graft was required. The plates had to be removed two months

The flap fully survived and the postoperative course was unevent-

after this surgery due to osteomyelitis. An external fixator was then

ful, with primary healing of the bone and soft tissues (Figure 4). At

placed and a radius Masquellet procedure was performed. As a result

5-month follow-up, the skeleton was healed. There was minimal osteo-

of an infected pseudarthrosis of the radius the patient underwent an

penia at the distal radial segment. Wrist extension was 48 degrees,

iliac cancellous bone graft. After removal of the external fixator a pos-

flexion 24 degrees, and pronation–supination was 58–0–48 degrees,

terior plaster forearm splint was placed.

with full finger flexion. The patient could hold a 4 kg dumbbell with the

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Shortening of the radius may result in ulnar abutment and wrist pain because of carpus impaction against the ulna, decreased grip strength, impingement of the DRUJ with loss of rotation, and radial inclination of the hand (George & Lawton, 2015; Patel & Paksima 2010). Various surgical procedures have been proposed for treatment of radial club hand, such as soft tissue releases with or without ulnar osteotomy, bone grafting, arthodesis, centralization, radialization, and lengthening procedures (Ilizarov’s methods) (de Jong et al., 2012; Romana et al., 2015). Centralization remains one of the most common surgical procedures. It involves removing bones from the wrist so that the hand sits straight on the end of the ulna (placed in a slot within the wrist). Radialization involves moving the hand toward the ulnar border (A) Drawing of the defect after debridement. (B) Drawing of the reconstruction. An arthrodesis of the DRUJ was performed combined with an ulnar resection osteotomy proximal to the arthrodesis. A fibula osteoseptocutaneous flap (F) was used to reconstruct the diaphyseal defect of the radius. The skin island of the flap was used for monitoring and because of scar tissue release (S). (PV: peroneal vessels) FIGURE 2

of the forearm; in addition the extensor and flexor carpi radialis (FCR) tendons of the wrist are transferred to the ulnar side to weaken the forces of radial deviation, and so that the hand is balanced on the end of the ulna. It is less likely to interfere with growth because less bone is resected and there is usually more mobility in the wrist because the muscles are restored and the ulna is not placed in a slot within the wrist.

elbow flexed without discomfort. No donor site-related complaints were reported by the patient, despite some weakness on ambulation for 3 months after surgery. His DASH score—Disabilities of the Arm, Shoulder, and Hand

Resection and replacement with vascularized bone may be indicated in subacute injuries for reconstruction of sizable defects and to restore bone alignment. Free fibula transfer has been extensively reported for long bones reconstruction, especially in the lower limb. In the upper limb, the forearm bones have similar cross-section compared

Questionnaire was 15.83 (It ranges from 0- no disability, to 100- most

to the fibula, and reconstruction of segmental defects of both radius

severe disability). The patient was very satisfied with the functional

and ulna with microvascularized fibula, or (less frequently) using a

results and didn’t complain about the receptor/donor site appearance

double-barrel free fibular flap have also been reported (Cavadas & Thi-

and scarring.

one, 2014; Safoury, 2005; Saint-Cyr, Farkas, & Gupta, 2008). A metatarsal head free flap was also described for distal ulna reconstruction

3 | DISCUSSION

and DRUJ stabilization (Cavadas & Thione, 2014). If DRUJ congruity is not possible, salvage techniques such as the

In open fractures, with soft tissue damage, bone loss, or severe commi-

Darrach procedure, or arthroplasty may be indicated (Lluch, 2010,

nution, external fixation is indicated to avoid further devascularization

2013; Yu et al., 2015). Arthrodesis is the most reliable and durable sur-

of the fragments (Helber & Ulrich, 2000). Among the most frequent

gical procedure for the treatment of a joint disorder, and its only disad-

complications of forearm fractures with associated DRUJ disruption

vantage is the loss of motion of the fused joint. The S-K technique

we find infection, nerve injury, limited range of motion (ROM), chronic

involves a combined arthrodesis of the DRUJ (between the ulnar head

instability of the DRUJ, and persistent pain (George & Lawton, 2015;

and sigmoid fossa of the distal radius) and an ulnar resection osteot-

Patel & Paksima 2010). Important bone loss, delayed union, mal-union,

omy proximal to the arthrodesis in order to maintain, or even improve

and non-union are also complications that may result in additional

forearm pronation and supination by creating a pseudoarthrosis of the

deformities (George & Lawton, 2015; Vanheest, 2006).

ulna (Lluch, 2013). It differs from the Darrach procedure in that it

FIGURE 3

(A) Flap markings. (B) Intraoperative image of flap after insetting; the skin island was placed externally

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Result after 5 months: (A) dorsal view; (B) radial aspect; (C) patient was holding a 4 kg dumbbell with the elbow flexed without discomfort; acceptable pronation–supination with good transverse stability of the forearm upon resisted elbow flexion. (D) anteroposterior; (E) lateral view: Postoperative X-ray at 5 months, showing well healed osteotomies. There were minimal degenerative changes in the distal radial segment

FIGURE 4

preserves ulnar support of the wrist, since distal radioulnar and ulnocar-

wide as 25 cm in the limbs. Furthermore, fibula is not essential for

 pez et al., 2013; Lluch, 2013; Yu pal ligaments are mantained (García-Lo

load-bearing and ambulation. In our clinical case, it was used to restore

et al., 2015). Aesthetic appearance is also superior after the S-K proce-

the anatomy of the altered segment acting as a bone substitute of

dure, as the normal prominence of the ulnar head, most noticeable when

adequate length, customized to the defect, covered by vascularized

the forearm is in pronation, is preserved.This procedure may be used in a

muscle, achieving bone union and gaining a satisfactory functional

variety of conditions altering the normal function of the ulnocarpal and

recovery. One skin island was added for coverage of osteosynthesis

DRUJ, such as irreducible DRUJ dislocations or ulnocarpal impaction. It

material, soft tissue reconstruction, and flap monitoring.

may be important when there is low bone stock of the distal ulna, insta-

Vascularized bone is highly successful in achieving union, but it is not

bility of the interosseous ligament of the forearm associated with an

risk-free, leading to complications like insufficient consolidation, stress

absence of radial head, or if an active infection or another soft tissue

fractures, and sometimes limb length discrepancy. In our clinical situation,

problem is present in the surgical site (Lluch, 2010, 2013). However, the

the S-K procedure was performed in order to improve stability of the

S-K procedure is not free of possible complications, such as nonunion or

DRUJ, which becomes a more stable platform on which the wrist may bal-

delayed union of the arthrodesis, fibrous or osseous union at the pseu-

ance, allowing the forearm to rotate into pronation and supination. Szabo,

doarthrosis, and painful instability at the proximal ulna stump, which can

Anderson, and Chen (2006) reported a procedure in which a distal radial

be prevented if a careful surgical technique is used (Lluch, 2013).

osteochondral allograft was combined with DRUJ arthrodesis and S-

Isolated ulna or radius shortening may also be reconstructed with

Kprocedure. They concluded that the addition of the S-K procedure

distraction osteogenesis by using a unilateral external fixator (Koca,

afforded functional stable wrist motion and decreased late collapse of the

Akpancar, & Yıldız, 2015). However, it is difficult to reach the length of

allograft. Other authors have compared the outcomes of osteoarticular

the normal side, pin breakage and pin tract infection can occur, defor-

allograft reconstruction in patients with and without the S-K procedure

mity or shortening may recur after the first time of distraction and may

(Li, Jiao, Guo, Ji, & Wang, 2015). Early narrowing of the joint and other

need repetitive osteotomy and distraction osteogenesis surgery (Koca

radiological signs of degenerative changes were more evident and severe

et al., 2015; Romana et al., 2015).

in patients without the S-K procedure as opposed to those with the addi-

Combined free fibular osteoseptocutaneous flap and S-K proce-

tional S-K procedure. It was postulated that instability of the DRUJ accel-

dure were used in this case for reconstruction of a radial club hand

erates the degenerative changes of the wrist. Grip strength and the range

restoring radius alignment in the context of pseudarthrosis of the

of pronosupination were also significantly higher in the patients with the

radius. Bone grafts yield high complication rates when gaps >6 cm

S-K procedure. However, to the best of our knowledge, there are no

must be restored. The structural characteristics of the fibula, strength,

reports regarding concomitant use of a free fibular osteoseptocutaneous

shape, length, and limited donor site morbidity make it an excellent

flap and S-K procedure for reconstruction of a postraumatic radial club

option for reconstructing long bone defects larger than 6 cm and as

hand.

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In summary, en bloc resection of the diseased bone till healthy margins are achieved clinically, adequate soft tissue release and reconstruction with a free fibular osteoseptocutaneous flap are an acceptable treatment for postraumatic radial club hand with good midterm results. The addition of the S-K procedure for reconstruction of the distal radius could improve functional outcomes and decrease the severity of degenerative changes of the wrist. Good results were achieved in this case and combined free fibular osteoseptocutaneous flap and S-K procedure may be considered in severe forms of posttraumatic radial club hand, however, further data

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Helber, M. U., & Ulrich, C. (2000). External fixation in forearm shaft fractures. Injury, 31, 45–47. Koca, K., Akpancar, S., & Yıldız, C. (2015). Correction of length discrepancy of radius and ulna with distraction osteogenesis: Three cases. Case Reports in Orthopedics, 2015, 656542. Li, J., Jiao, Y., Guo, Z., Ji, C. & Wang, Z. (2015). Comparison of osteoar-Kapandji ticular allograft reconstruction with and without the Sauve procedure following tumour resection in distal radius. Journal of Plastic, Reconstructive & Aesthetic Surgery, 68, 995–1002 –Kapandji procedure: Indications and tips for Lluch, A. (2010). The Sauve surgical success. Hand Clinics, 26, 559–572.

are necessary.

–Kapandji procedure. Journal of Wrist Surgery, Lluch, A. (2013). The Sauve 2, 33–40. Feb;

CONFLICT OF INTEREST

Patel, V. P., & Paksima, N. (2010). Complications of distal radius fracture fixation. Bulletin of the NYU Hospital for Joint Diseases, 68, 112–118.

All named authors hereby declare that they have no conflicts of interest to disclose.

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Romana, C., Ciais, G., & Fitoussi, F. (2015). Treatment of severe radial club hand by distraction using an articulated mini-rail fixator and transfixing pins. Orthopaedics & Traumatology: Surgery & Research, 101, 495–500. Safoury, Y. (2005). Free vascularized fibula for the treatment of traumatic bone defects and nonunion of the forearm bones. Journal of Hand Surgery [British], 30, 67–72. Saint-Cyr, M., Farkas, J., & Gupta, A. (2008). Double-barrel free fibula flap for treatment of infected nonunion of both forearm bones. Journal of Reconstructive Microsurgery, 24, 583–587. Szabo, R. M., Anderson, K. A., & Chen, J. L. (2006). Functional outcome of en bloc excision and osteoarticular allograft replacement with the Sauve–Kapandji procedure for Campanacci grade 3 giant-cell tumor of the distal radius. Journal of Hand Surgery America, 31, 1340–1348. Vanheest, A. (2006). Wrist deformities after fracture. Hand Clinics, 22, 113–120. Yu, Y. D., Wu, T., Tian, F. T., Shang, Y. T., Yu, X. F., Bai, Y. B., & Han, C. L. (2015). Ulnar impaction syndrome with different operative methods: A comparative biomechanical study. International Journal of Clinical and Experimental Medicine, 8, 5715–5722.