Bilateral Kienböck’s Disease

INTRODUCTION

Kienböck’s disease usually affects only one wrist, most commonly the dominant one (Kristensen et al., 1986). The incidence of bilateral Kienböck’s disease is very low and there are only case reports and short series of such cases (Edmunds and Harvey, 1998; Kahn and Bade, 1986; Lin et al., 1983; Mok et al., 1997, Morgan and McCue, 1983; Steinhäuser and Posival, 1982; Taniguchi and Tamaki, 1998). This paper describes and compares the clinical and radiographic features of 11 patients with bilateral Kienböck’s disease with those of patients with unilateral Kienböck’s disease in order to see if this demonstrates any factors important in the aetiology of the condition.

PATIENTS AND METHODS

Between 1969 and 2001 (33 years), 251 patients with Kienböck’s disease were treated at our hospital. Of these, 161 patients (64%) had right wrist disease, 79 (32%) had left wrist disease and 11 patients (4%) had bilateral Kienböck’s disease. The medical records of these 11 patients were reviewed and the radiographs taken at their first clinical appointment were assessed. A baseline on the posteroanterior view was drawn along the middle of the shaft of the ulna and the following measurements were made: ulnar variance, radial inclination angle, carpal height ratio and carpal ulnar distance ratio. The radiolunate angle (RLA), radio-scaphoid angle, scapholunate angle and Ståhl’s index were measured on the lateral films.

For the purpose of comparison, medical records and radiographs of all 31 patients presented with unilateral Kienböck’s disease at our hospital between 1997 and 2000 were also reviewed. There were 21 men and 10 women, with a mean age of 39 (range 17–73) years. In addition, 33 normal wrist radiographs were reviewed. These were from 10 men and 21 women, with a mean age of 47 (range 20–63) years.

Finally, we compared the radiographs of both wrists of the patients with bilateral and unilateral Kienböck’s disease.

Statistical analyses were performed using the unpaired t-test and the Mann–Whitney U-test.

RESULTS

Demographics, radiographic findings, treatments and occupations/activities of the 11 patients with bilateral Kienböck’s disease are summarized in Table 1. There were 10 men and one woman, with a mean age of 40 (range 14–57) years.

Diagnoses were based on radiographic findings. Unfortunately, the pre-operative radiographs of the left wrist of Case 1 had been lost and the right wrist of Case 3 had been treated by excision of the lunate at another hospital. At the first clinical appointment, radiographs of 19 of the 20 wrists showed flattening of the lunate. The radiographs of the other wrist showed generalized increased density of the lunate, which is characteristic of Kienböck’s disease. One wrist was classified as Lichtman stage II, six as stage IIIA, 12 as stage IIIB and one as stage IV (20 wrists).

Eight of the 11 patients were engaged in manual work that required repetitive motion of both hands. Most of their occupations were heavy-lifting in nature and two patients were athletes. None of the patients had ever been prescribed corticosteroids, and none had any general predisposing factors for avascular necrosis. No patient had avascular necrosis at any other site. One of the 11 patients reported a history of injury predating the exacerbation of symptoms.

Four of the 11 patients had symptoms in both wrists and two of these four patients had undergone surgical treatment in both wrists, while the other two had undergone conservative treatment of both wrists. The intervals between the onset of the symptoms in one wrist and the other ranged from 17 years (Case 1) to 4 months (Case 11). Seven of the 11 patients predominantly had symptoms in only one wrist, with one of them only having had mild symptoms for a short period of time in the other wrist. These seven cases needed no treatment for the asymptomatic wrist. The five of the seven wrists without significant symptoms were non-dominant (one stage II, four stage IIIB) and the other two were dominant (two stage IIIA) (Fig 1).

Of 31 patients with unilateral Kienböck’s disease, one wrist was assessed as Lichtman’s stage II, 15 as stage IIIA, 12 as stage IIIB, and three as stage IV. Twenty of the 31 (65%) were engaged in manual work and two patients were athletes, both tennis players. No patients reported a history of trauma.

The radiographic parameters of the patients with bilateral Kienböck’s disease were compared to those of the unilateral patients. There were no statistically significant differences in any of the radiographic parameters (Table 2).

Comparison of the radiographs of the normal patients with those of the patients with bilateral Kienböck’s disease revealed significant differences in RLA and Ståhl’s index, but no other differences.

We compared the radiological measurements on the normal wrists of our 31 patients with unilateral Kienböck’s disease with the same measurements from the contralateral side which was involved in Kienböck’s disease. There were no statistical significant differences between these measurements including ulnar variance. We also compared the difference between two sides of our nine cases of bilateral Kienböck’s disease, but could not find any differences.

DISCUSSION

We found 24 reports of bilateral Kienböck’s disease: Steinhäuser and Posival (1982) reported six, Taniguchi and Tamaki (1998) five, Kristensen et al. (1986) three, Rasmussen and Schantz (1987) three, Morgan and McCue (1983) two and Lin et al. (1983) two cases of bilateral Kienbock’s disease. Furthermore, there are three single case reports of bilateral disease (Edmunds and Harvey, 1998; Kahn and Bade, 1986; Mok et al., 1997). The occupations of 14 patients were described in four of these nine reports and nine of these were engaged in manual work. Three patients had a collagen disease (two SLE and one lupoid hepatitis), and two of these had had corticosteroid treatment.

The reported incidence of bilateral Kienböck’s disease appears fairly consistent, at 3–7% (Table 3). We could not find any radiographic parameters, including ulnar variance, that were risk factors for bilateral, rather than unilateral, Kienböck’s disease.

In aseptic necrosis of the femoral head, corticosteroids and/or systemic lupus erythematosus (SLE) are recognized as risk factors. All Japanese epidemiologic research has shown that 55% of patients with osteonecrosis of the femoral head have taken corticosteroids and 37% have SLE. In contrast only one of our 251 patients with Kienböck’s disease and none with bilateral Kienböck’s disease had taken corticosteroids or been diagnosed with a collagen disease. In previous reports of bilateral Kienböck’s disease, Taniguchi and Tamaki (1998) reported on two patients with corticosteroid treatment, of which one had SLE. Mok et al. (1997) described one patient with SLE who had taken corticosteroids. In cases of unilateral Kienböck’s disease, four patients with SLE, who was treated with corticosteroids developed Kienböck’s disease (Griffiths et al., 1979; Labowitz and Schumacher, 1971; Lightfoot and Lotke, 1972; Urman et al., 1977). Corticosteroids may generate fat emboli, which may compromise the blood supply to the lunate, and SLE vasculitis may also compromise its blood supply but they do not appear to be a risk factor for bilateral, rather than unilateral, Kienböck’s disease.

Kristensen et al. (1986) reported that 39 of 46 patients (85%) with Kienböck’s disease were engaged in hard physical work and Rasmussen and Schantz (1987) reported that 80 of 93 patients (86%) were heavy manual labourer. Nakamura et al. (1991) reported that 82 of 122 patients (67%) were manual workers without any history of trauma. Repetitive stress associated with manual labour such as hammering, may contribute to the development of Kienböck’s disease but no case control study has ever been performed to confirm or refute the opinion. In the present study, 65% of our patients with unilateral Kienböck’s disease and eight of the 11 patients with bilateral disease were manual workers. In previous reports of bilateral Kienböck’s disease, which contained descriptions of occupation, nine of 14 patients were manual labourers (Edmunds and Harvey, 1998; Morgan and McCue, 1983; Steinhäuser and Posival, 1982; Taniguchi and Tamaki, 1998).

Our patients with bilateral Kienböck’s disease did not always present themselves with pain in both wrists. Nine dominant wrists and seven non-dominant wrists were symptomatic. Stage IIIB diseases did not always cause symptoms in non-dominant wrists, suggesting that mechanical stress contributes to the development of symptoms. When patients with unilateral wrist pain come to our hospital, we routinely take radiographs of both wrists, which increases the chance of diagnosing bilateral Kienböck’s disease.