Intraarticular osteotomy for adult Madelung deformity: Case report

Abstract

Madelung deformity is a hemi-epiphyseal dysplasia of the radioulnar axis. The prominent feature is radial deformity secondary to premature closure of the volar-ulnar side of the distal radial physics. The distal radius is malaligned with excessive ulnar and volar tilt, shortening and concomitant ulna plus deformity.

Adults presenting most frequently complain of pain, limited wrist motion and poor cosmesis. Surgical indications in adult patients are severe pain and/or limitation of wrist motion. Current surgical treatments consist of various types of extraarticular osteotomies to improve the alignment of the distal radius epiphysis, without addressing the intrinsic deformity of the articular surface itself.

The authors present a novel technique of intraarticular osteotomy in an adult patient with severely deformed articular distal radial articular facets. To the best of the authors’ knowledge, no similar technique has been previously described in the literature.

According to the Oberg-Manske-Tonkin (OMT) classification of congenital hand differences, Madelung deformity is a hemi-epiphyseal dysplasia of the radioulnar axis.¹ It is more frequently seen in females, and it is commonly bilateral.² The prominent feature is radial deformity secondary to premature closure of the volar-ulnar side of the distal radial physis, although the etiology and pathomechanics remain obscure. The distal radius is malaligned with excessive ulnar and volar tilt, shortening and concomitant ulna plus deformity. In severe cases, usually associated with Lery Weil dyschondrosteosis, radial bowing is present.³

Adults presenting with Madelung deformity most frequently complain of pain, followed in frequency by limited wrist motion and poor cosmesis. Surgical indications in adult patients with Madelung deformity are severe pain and or limitation of wrist motion.⁴ Current surgical treatments consist of various types of extraarticular osteotomies to improve the alignment of the distal radius epiphysis,³ without addressing the intrinsic deformity of the articular surface itself.

The authors present a novel technique of intraarticular osteotomy in a bilateral adult patient, with severely deformed articular distal radial articular facets. To the best of the authors’ knowledge, no similar technique has been previously described in the literature.

Clinical case

A 67-year-old female patient presented with a bilateral Madelung deformity of the wrists, with severe pain (visual analogue scale (VAS 6) and reduced active range of motion (AROM) (Table 1). Radiographic assessment followed the guidelines suggested by McCarroll et al.⁵ and included the lunate fossa angle (LFA), and ulnar tilt angle (UT). Lunate subsidence (LS) was not relevant because the planned Sauvé-Kapandji procedure. Preoperative values are summarized in Table 1. Severe ulna plus and DRUJ incongruence were also prominent features (Figure 1). Preoperative DASH score was 78.

Table 1.

Clinical and radiographic parameters of the patient.

	Preoperative	Postoperative at 12 months
AROM Extensión-flexion	Left 30°−0°−20° Right 20°−0°−20°	Left 50°−0°−30° Right 55°−0°−30°
AROM supination-pronation	Left 40°−0°−20° Right 50°−0°−30°	Left 80°−0°−80° Right 70°−0°−70°
VAS pain	Left and right 6	Left and right 1
LFA	Left 62° Right 68°	Left 40° Right 40°
UT	Left 46° Right 50°	Left 40° Right 40°

AROM, active range of motion; VAS, visual analogue scale; LFA, lunate fossa angle; UT, ulnar tilt angle.

Figure 1.

Preoperative X-ray showing the deformity of the wrists. LFA indicated in red and UT in yellow are measured as the complementary angle between the corresponding red and yellow lines and the blue ulnar axis, according to Mc Carroll.⁵

Due to the gross coronal-plane convexity of the distal radius articular surface, the already described extraarticular osteotomies were deemed inadequate to correct the deformity in this patient. A self-retaining intraarticular osteotomy was planned to correct the lunate fossa angle and render a concave distal radial articular surface (Figure 2). In the posteroanterior Xray, the ideal profile of the lunate fossa was drawn, and the length of the actual lunate fossa (distance between points A and B) was marked (A–C). The distance between points B and C and the angle ACB were measured. With these parameters a triangle was drawn (ABD) with the same angle (ACB = ABD) and slightly larger (BD 2 mm longer than CB). This is the osteotomy segment to be rotated to reposition the lunate fossa.

Figure 2.

Depiction of the surgical procedure. The ideal profile of the lunate fossa is marked, and the distance AB is marked in this line, creating point C. The distance CB and the angle ACB are measured. Point D is marked in the radial metaphysis, so the distance BD is 2 mm longer than CB and the angle ABD is similar to ACB. Then the triangle ABD is cut and rotated to the optimal lunate fossa position and a Sauvé-Kapandji procedure is associated, using the resected ulnar neck as bone graft for the resultant metaphyseal defect of the radius.

Through a volar approach the triangular osteotomy of the lunate fossa was performed as planned, and the segment rotated. The extra 2 mm-length of the triangular segment allows bone contact between the apex of the osteotomized segment and the ulnarmost cortex of the distal radius. A Sauvé-Kapandji procedure was associated, and the resected segment of the ulna was used as local bone graft (Figure 3). Fixation was performed with 1.4 mm K wires because of the poor quality of the bone. Plate fixation would be preferable. A short arm plaster splint was worn for 6 weeks, allowing pronation supination. K wires were removed at 8 weeks, and ROM exercises were allowed afterwards.

Figure 3.

Intraoperative view of the osteotomized and rotated lunate fossa.

The left wrist was operated on first, and the right wrist was corrected three months later.

The radiographic and clinical results at 12 months are summarized in Table 1, with significant improvement in all parameters.

There was one complication in the right-hand surgery. Bone regrowth at the Sauvé-Kapandji resection focus occurred, along with nonunion of the distal radioulnar fusion, blocking pronation supination. Surgical removal of the bone and revision of the radioulnar fixation solved the problem.

12 months postoperative DASH score was 21.

Discussion

Madelung deformity in the adult patient is usually diagnosed because of wrist pain, decreased motion or wrist deformity. Severe pain due to gross malalignment of the distal radial epiphysis or ulnocarpal impaction requires surgical correction. A number of distal metaphyseal osteotomies have been described, the dome osteotomy being the most popular because of its versatility and lack of resultant bone shortening.⁶ More recently the virtual planning and 3D printed cutting guides technology has allowed a more precise execution of these osteotomies.^7,8

In severe cases, the distal radial epiphysis itself is grossly deformed, with increasing LFA, resulting in a convex distal radial articular surface, as in the case presented. With this severe coronal-plane convexity, the described extraarticular osteotomies would only partially address the deformity.

The design of the lunate fossa osteotomy presented herein, as an inverted pyramid segment, allows coronal rotation and self-retaining proximal bone contact. The metaphyseal bone defect created was filled with local bone graft from the resected distal ulna. Because of gross ulna plus deformity with incongruent DRUJ, a Sauvé-Kapandji procedure was included in the correction. Internal fixation was initially planned, but the poor quality of the bone stock of the distal radius made percutaneous fixation with K-wires more advisable.

The clinical and radiological improvement was remarkable (Table 1), with resolution of the pain. Remarkably, the postoperative LFA and UT were similar, indicating correction of the radiocarpal alignment. The complication that occurred in the left hand (partial reossification at the Sauvé-Kapandji resection) could be easily corrected with an additional minor surgery and was not directly related to the intraarticular radial osteotomy procedure.

The technique presented is relatively straightforward to plan and perform through a standard volar approach. Although it is a single case report and more cases are needed to fully assess its potential, the radiographic and clinical improvement are encouraging. This technique could be an option in adult Madelung deformities with severe convex deformity and high LFA.

Declaration of competing interest

The authors do not have any potential conflict of interest with respect to this manuscript.