Abstract
Objective To evaluate the outcomes and complication rate of surgical management in adolescent patients with Kienböck's disease and compare lunate offloading and revascularization procedures.
Methods We performed a retrospective chart review to evaluate adolescent patients with Kienböck's disease between 1990 and 2016 who were surgically managed. Charts were reviewed for demographic information, presence of trauma, range of motion, grip strength, and radiographic parameters pre- and postoperative.
Results We assessed 21 wrists in 20 patients. All had failed conservative management and required surgery. Seven patients underwent lunate offloading procedures, most commonly radial-shortening osteotomy, whereas 13 patients had an attempt at revascularization. All patients had either minimal or no pain at a clinical mean clinical follow-up of 63.4 months. Postoperatively, grip strength and radial deviation improved, with no difference between the two groups. Those that underwent joint offloading procedures had less ulnar variance. Eight of 11 patients with a postoperative MRI (magnetic resonance imaging) had evidence of lunate revascularization after a revascularization procedure.
Conclusion Surgical management of Kienböck's disease in adolescent patients can yield satisfactory outcomes in those that fail conservative management.
Level of Evidence/Type of study This is a Level IV, therapeutic study.
Keywords: Kienböck's disease, adolescent Kienböck's disease, joint offloading, revascularization, bone graft
Kienböck's disease is an idiopathic avascular necrosis affecting the lunate. It can lead to lunate fragmentation with carpal collapse and eventual perilunate arthritic changes. 1 2 Commonly, patients present with wrist pain, swelling, decreased grip strength, and wrist motion. Many factors have been suggested to contribute to the development of Kienböck's disease, such as the length of the ulna, vascularity of the lunate, morphology of the lunate, previous trauma, and amount of radial inclination. 3 Most commonly, patients present between 20 and 40 years of age. 2 4 It is rare in adolescent or skeletally immature patients, of which there is currently no gold standard of treatment. 5
Lichtman et al presented an updated algorithm for the management of Kienböck's disease. They felt that those patients under 15 years of age should undergo nonoperative management. However, those patients still symptomatic or with radiographic progression after 3 to 6 months with Lichtman stage 0 to II Kienböck's disease should be offered surgery. 6 Procedures can be grouped into those that aim to unload or decompress the lunate to prevent fragmentation and progression to carpal arthritis and those that revascularize or reconstruct the lunate. Ferlic et al summarized the current literature on Kienböck's disease in adolescent patients, which consisted mostly of case reports and small case series. 2 The long-term outcomes of surgery are unknown, whereas some studies suggest that younger patients have better results due to increased remodeling potential and that younger age is an important prognostic factor. 5 7 The majority of Kienböck patients were treated with either nonoperative casting or radial-shortening osteotomy to offload the lunate. 2
Our goal was to analyze our outcomes of adolescent patients presenting with Kienböck's disease who failed conservative management and to describe the surgical management and outcomes at our institution, particularly the role of revascularization versus lunate offloading procedures.
Methods
Following Institutional Review Board approval, we retrospectively identified patients under 18 years of age with radiographic evidence of Kienböck's disease who were treated at one institution between 1990 and 2016. Choice of procedure was based on ulnar variance and surgeon preference. We excluded those with less than 12 months of clinical follow-up and those with previous surgery. Charts were reviewed for patient demographics, pre- and postsurgical physical findings, timing of surgery, and complications. In addition, we obtained clinical follow-up data with special emphasis on functional outcomes during the last noted clinical follow-up. This included resolution of pain, range of motion (ROM), grip strength (measured using Jamar dynamometer, Therapeutic Equipment Corporation, Clifton, NJ), the Mayo Wrist Score (MWS), the Disabilities of the Arm, Shoulder, and Hand Questionnaire (DASH), and the Patient-Rated Wrist Evaluation (PWRE).
Pre- and postoperative radiographic evaluation with roentograms, computed tomography (CT), and magnetic resonance imaging (MRI) was performed to look at ulnar variance, Lichtman staging, 1 presence of avascular necrosis, and progression of disease. Radiographic findings consistent with Kienböck's disease include lunate sclerosis, fracture lines, subchondral cysts, and fragmentation and collapse with eventual arthrosis and scapholunate dissociation. On MRI, ischemic marrow leads to a high signal on T2-weighted images that then changes to a low signal as the lunate becomes necrotic. 1 Carpal height (as per Youm et al 8 ), Stahl's lunate index, 9 and radioscaphoid angle (RSA) were measured by two independent observers pre- and postoperatively to assess the severity of Kienböck's disease.
Statistical Analysis
The data were summarized using means and standard deviations (SDs) for continuous variables and using counts and percentages for categorical variables. Outcomes comprising continuous variables such as pre- and postoperative DASH scores were compared between the groups using Wilcoxon's signed rank test, whereas categorical variables were evaluated using chi-square tests (Kruskal–Wallis test if more than two independent variables and Mann–Whitney U test if two or less). Spearman correlation coefficient was used for correlation analysis. All statistical tests were two-sided, and p < 0.05 was considered significant.
Results
Our search revealed 21 wrists in 20 patients ( Table 1 ). All patients presented with wrist pain and decreased ROM. Mean patients' age at surgery was 16.7 years (range: 13–18 years), and follow-up time was 63.4 months (range: 12–252 months). Mean radiographic follow-up was 43.8 months (1–208 months). One patient had a past history of prednisone use. Time from symptom onset to surgery was 12.1 months (mean: 10.0 months; range: 3–36 months). Over half of the wrists had a history of previous trauma (12/21 wrists), and fall on an outstretched hand was the most common mechanism of injury. All patients underwent a trial of casting at an outside institution for 3 months without resolution of symptoms.
Table 1. Characteristics of pediatric patients undergoing surgery for Kienböck's disease.
| Number of patients (%) | |
|---|---|
| Sex | |
| Male | 11 (55) |
| Female | 9 (45) |
| Age, years (mean: 16.7; range: 13–18; SD: 1.6) | |
| ≤15 | 6 (30) |
| > 15 | 14 (70) |
| Side | |
| Right | 13 (61.9) |
| Left | 8 (38.1) |
| Dominant involved | |
| Yes | 12 (57.1) |
| No | 9 (42.9) |
| Lichtman staging | |
| Preoperative ( n = 19) | |
| Stage II | 2 (9.5) |
| Stage IIIA | 13 (61.9) |
| Stage IIIB | 4 (21.1) |
| Postoperative ( n = 17) | |
| Stage II | 1 (5.8) |
| Stage IIIA | 12 (70.6) |
| Stage IIIB | 4 (23.5) |
| DASH | |
| Postoperative ( n = 14) | 8.8 (SD: 11.0) |
| MWS | |
| Postoperative ( n = 11) | 77.7 (SD: 8.8) |
| PRWE | |
| Postoperative ( n = 13) | 10.7 (SD: 14.3) |
Abbreviations: DASH, Disabilities of the Arm, Shoulder, and Hand Questionnaire; MWS, Mayo Wrist Score; PRWE, Patient-Rated Wrist Evaluation; SD, standard deviation.
In our series, 8 wrists underwent lunate offloading procedures, 13 underwent a vascularized bone grafting (VBG), and 1 wrist underwent a combined procedure ( Table 2 ). At the last follow-up, all wrists had either no or minimal pain. There was a significant increase in grip strength and radial deviation postoperatively ( Table 3 ). There was no difference in ROM when comparing vascularized bone reconstructions with offloading procedures and combined procedures. The average DASH, MWS, and PWRE scores were 8.8, 77.7, and 10.7, respectively, postoperatively. Two (18%) patients were excellent, three (27%) were good, and six (54%) were satisfactory based on the MWS.
Table 2. Surgical procedures for the management of adolescent Kienböck's disease in our patient series.
| Procedure | Number of patients |
|---|---|
| Lunate offloading alone | |
| Radial shortening | 5 |
| Ulnar lengthening | 1 |
| Scaphocapitate fusion | 1 |
| Vascularized bone graft alone | |
| 4 + 5 ECA | 8 |
| Second IMCA | 2 |
| Medial femoral condyle + lunatectomy + midcarpal fusion | 1 |
| Radial VBG based on retrograde flow through PIA | 2 |
| Combined | |
| Radial shortening and VBG using retrograde flow through PIA | 1 |
| Concomitant procedures | |
| PIN or AIN neurectomy | 4 |
| External fixator alone | 3 |
| Scaphocapitate pin alone | 4 |
| Radioscaphoid pin plus fixator | 4 |
| Scaphocapitate pin plus fixator | 1 |
Abbreviations: AIN, anterior interosseous nerve; ECA, extensor compartment artery; IMCA, intermetacarpal artery; PIA, posterior interosseous artery; PIN, posterior interosseous nerve; VBG, vascularized bone grafting.
Table 3. Pre- and postoperative ROM for adolescent patients with surgical management of Kienböck's disease.
| Preoperative ROM (degrees) | Contralateral wrist (%) | Last clinical follow-up, ROM (degrees) | Contralateral wrist (%) | p -Value a | |||||
|---|---|---|---|---|---|---|---|---|---|
| N | Mean | SD | N | Mean | SD | ||||
| Flexion | 20 | 39.4 | 13.4 | 52.0 | 20 | 47.5 | 15.3 | 64.6 | 0.185 |
| Extension | 20 | 45.7 | 11.9 | 64.2 | 20 | 48.3 | 14.2 | 74.3 | 0.351 |
| Radial deviation | 19 | 14.2 | 8.1 | 58.2 | 17 | 20.7 | 7.2 | 71.4 | <0.05 |
| Ulnar deviation | 19 | 32.9 | 12.2 | 76.2 | 17 | 33.9 | 9.8 | 72.1 | 0.897 |
| Grip strength (kg) | 20 | 19.5 | 11.8 | 55.2 | 17 | 29.0 | 13.4 | 91.5 | <0.05 |
| Appositional strength (kg) | 6 | 9.2 | 7.5 | 98.4 | – | ||||
| Oppositional strength (kg) | 4 | 8.6 | 4.9 | 93.2 | – | ||||
| Pronation | 4 | 83.8 | 7.5 | 94.5 | – | ||||
| Supination | 4 | 88.7 | 8.1 | 95.0 | – | ||||
Abbreviations: ROM, range of motion; SD, standard deviation.
Wilcoxon's signed rank test.
Radiographically, the average preoperative ulnar variance was –1.41 mm. There were 13 wrists with negative ulnar variance, 1 with neutral ulnar variance, and 3 with positive ulnar variance. Preoperatively, 2 wrists were Lichtman stage II, 13 were IIIA, and 4 were IIIB. The average postoperative ulnar variance was –0.79 ( Table 4 ). There was no significant difference between pre- and postoperative ulnar variance when comparing all patients. Postoperatively, 1 wrist was Lichtman stage II, 12 wrists were IIIA, and 4 wrists were IIIB. Two patients with stage II and stage IIIA, respectively, did not have long-term follow-up radiographs to allow proper staging postoperatively. No wrists downgraded their staging postoperatively. There was no difference in pre- and postoperative staging. There were 11 wrists that underwent postoperative MRI, and 8 of those had evidence of revascularization. All of these wrists had undergone VBG. Of the three who did not have evidence of revascularization, two were after a 4 + 5 extensor compartment artery and the third was after a second metacarpal base VBG.
Table 4. Radiographic parameters after surgical management of Kienböck's disease in an adolescent population.
| Preoperative | Postoperative | Delta ( n = 16) | p -Value a | |
|---|---|---|---|---|
| Ulnar variance ( n = 16) | –1.41 (SD: 2.1) | –0.79 (SD: 1.8) | 0.54 (SD: 2.8) | 0.498 |
| Carpal height ratio ( n = 15) | 0.48 (SD: 0.03) | 0.50 (SD: 0.1) | 0.038 (SD: 0.1) | 0.345 |
| Stahl's index ( n = 14) | 0.36 (SD: 0.1) | 0.35 (SD: 0.1) | –0.01 (SD: 0.1) | 0.917 |
| Radioscaphoid angle ( n = 10) | 46.7 (SD: 12.2) | 41.61 (SD: 17.7) | 8.13 (SD: 22.6) | 0.463 |
Wilcoxon's signed rank test.
When comparing lunate offloading procedures to VBGs, there was no difference in incidence of trauma, hand dominance, time from symptom onset to surgery, ROM, or grip strength pre- or postoperative, presence of avascular necrosis preoperatively, preoperative ulnar variance, and Stahl's index pre- or postoperative ( Table 5 ). Lunate offloading procedures had a ratio change of 0.16, whereas the VBG's carpal height ratio only changed by 0.02, which trended toward significance ( p = 0.053). The postoperative ulnar variance was significantly less in the offloading procedures, as expected. There was no difference in clinical or radiographic outcomes when stratified based on Lichtman stage pre- or postoperatively. There was no difference in patient-related outcomes based on reconstruction type.
Table 5. Difference in parameters between lunate offloading procedures, vascularized bone grafting, and combined procedures.
| Lunate offloading (7) | Vascularized grafting (13) | Combined (1) | p -Value | |
|---|---|---|---|---|
| Age (years) | 17.1 | 16.4 | 16.7 | 0.414 |
| Dominant hand involved | 4 | 7 | 1 | 0.681 |
| Previous trauma | 6 | 6 | 0 | 0.129 |
| Symptom duration (months) | 9.1 | 11.5 | 3.5 | 0.302 |
| Preoperative grip strength | 17.8 | 20.4 | 18.0 | 0.961 |
| Postoperative grip strength | 24.2 | 31.4 | 28.0 | 0.470 |
| Lichtman preoperative | 0.122 | |||
| Stage II | 1 | 1 | 0 | |
| Stage IIIA | 5 | 9 | 0 | |
| Stage IIIB | 0 | 3 | 1 | |
| Lichtman postoperative | 0.317 | |||
| Stage II | 0 | 1 | 0 | |
| Stage IIIA | 5 | 8 | 0 | |
| Stage IIIB | 1 | 3 | 1 | |
| Radiographic parameters | ||||
| Preoperative UV | –1.2 | –1.4 | –1.4 | 0.944 |
| Postoperative UV | 0.0 | –2.2 | 2.5 | <0.05 a |
| Pre-CH | 0.51 | 0.47 | – | 0.225 |
| Post-CH | 0.58 | 0.47 | – | 0.425 |
| CH delta | 0.16 | 0.02 | – | 0.053 |
| Pre-Stahl | 0.35 | 0.36 | – | 0.796 |
| Post-Stahl | 0.30 | 0.37 | – | 0.315 |
| Stahl delta | 0.04 | –0.02 | – | 0.389 |
| Pre-RSA | 58.9 | 40.6 | – | <0.05 a |
| Post-RSA | 44.7 | 40.8 | – | 1.00 |
| RSA delta | 17.8 | 4.2 | – | 0.439 |
| Patient-reported outcomes postoperatively | ||||
| DASH | 6.8 | 10.4 | 1.7 | 0.600 |
| PWRE | 10.1 | 12.3 | 0.272 | |
| MWS | 80.0 | 77.5 | – | 0.552 |
Abbreviations: CH, carpal height; DASH, Disabilities of the Arm, Shoulder, and Hand Questionnaire; MWS, Mayo Wrist Score; PWRE, Patient-Rated Wrist Evaluation; RSA, radioscaphoid angle; UV, ulnar variance.
Wilcoxon's signed rank test.
Postoperatively, three patients had their hardware removed, two after radial shortening and one after an ulnar lengthening. One patient had transient median nerve paresthesias after a second metacarpal artery bone grafting. One patient had a nonunion after ulnar lengthening with iliac crest bone graft placement after 8 months and subsequent hardware removal after 3 months. One patient underwent a nonunion after radial shortening who then underwent iliac crest bone grafting and achieved union but subsequently required a wrist denervation. This would suggest ongoing wrist pain. Three patients had a pin-site infection from the external fixator, and one patient had a broken pin of the external fixator. Overall, 28% of patients who underwent a lunate offloading procedure had a complication versus 31% of those who underwent a revascularization procedure ( Table 6 ).
Table 6. Difference in complications between lunate offloading procedures, vascularized bone grafting, and combined procedures.
| Complication | Lunate offloading | Vascularized grafting |
|---|---|---|
| Hardware removal | 2 | 1 |
| Median nerve paresthesias | 1 | |
| Nonunion | 2 | |
| Pin-site infection | 3 | |
| Broken external fixator | 1 | |
| Total | 2/7 (28%) | 4/13 (31%) |
Discussion
Kienböck's disease is an uncommon disorder in skeletally immature patients. Typically, conservative measures are used; however, if patients have persistent pain and morbidity, then surgical options may be considered. There are multiple case reports of varying success with conservative and surgical methods. Hurley and McKee described a case of a 13-year-old female who presented with a stage IIIA Kienböck's disease 5 months after a hyperextension injury. She underwent a radial-shortening osteotomy, with resolution of symptoms at 8 months postoperatively. 4 Matsuhashi et al performed radial-shortening osteotomy on eight skeletally immature patients aged between 11 and 18 years, with all patients achieving excellent outcomes at 69 months. 10 They found revascularization of the lunate in six of eight of their patients and no advancement of Lichtman staging for those who obtained MRI follow-up. Ferlic et al provided a literature review and a case report of a 13-year-old with atraumatic wrist pain who was found on MRI to have stage III Kienböck's disease. 2 He underwent a radial-shortening osteotomy and subsequent lunate revascularization. Irisarri et al also provided a literature review of infantile and juvenile Kienböck's and described their experience of 13 cases. Their infantile patients responded to conservative means, whereas three of their nine juvenile patients underwent radial-shortening osteotomy with good clinical and radiological outcomes at 5 years. 11 Afshar presented two cases of Kienböck's disease in skeletally immature patients, one who had resolution of pain with casting and one who underwent radial-shortening osteotomy with improved symptoms. 5
The majority of adolescent patients who receive surgical management undergo lunate offloading procedures. 2 5 11 Other authors have attempted distal radius epiphysiodesis, temporary scaphotrapezoid joint fixation, capitate shortening, and ulnar lengthening to manage Kienböck's disease in skeletally immature patients with success. 5 There has only been one case series in which adolescent patients underwent a revascularization procedure. Kim et al reported on two patients who underwent radial shortening in conjunction with a VBG to the lunate. One patient was 15 and one was 14, both of whom were pain-free postoperatively. They did not obtain postoperative MRIs and did not comment on postoperative radiographic parameters. 12 We performed a VBG alone in 13 patients and a combined procedure in 1 further patient. All had minimal-to-no pain at the final clinical follow-up. Those with VBG alone had an improved carpal height ratio and RSA compared with the lunate offloading procedures alone. We had evidence of lunate revascularization in 8 of 11 patients who underwent an MRI postoperatively. Of the remaining three patients, one had questionable revascularization and two did not. They had good clinical outcomes, with one patient having no pain and two having minimal pain without any subsequent surgery.
Our series demonstrates 21 wrists with surgical management of Kienböck's disease who failed conservative casting or splinting. Overall, patients had increased grip strength, radial deviation, and excellent patient-reported outcomes. All patients had either no or minimal pain. We did not find a significant difference in Lichtman stage postoperatively despite excellent clinical outcomes. This is consistent with the cumulative reports of outcomes in skeletally immature patients. 5 There were no significant differences in clinical outcomes between those who underwent offloading versus revascularization procedures; however, radiographic parameters differed. Those who underwent offloading procedures had less ulnar variance postoperatively, as expected. The importance of ulnar variance, however, is not clear in skeletally immature patients. 5 Not all of our patients achieved revascularization of their lunate after a revascularization procedure; however, these patients had minimal-to-no pain postoperatively. Overall, the MWS demonstrated satisfactory outcomes, and DASH scores suggested minimal disability comparable to the general population.
We recognize the limitations of this study, including its retrospective and descriptive nature. The sample size was a sample of convenience, as it included all patients who presented for failed conservative management during our study period. There was a potential for recall bias, as the surveys were completed sometimes years after the original surgery. We were unable to obtain preoperative patient-related outcomes scores, and thus we were unable to assess a change after surgery. Ulnar variance has been suggested to be unreliable in skeletally immature patients due to its varied time of closure and incomplete ossification, and thus these outcomes may not be accurate. 13 Furthermore, because we were able to assess only the final outcome at the last follow-up, we are unable to follow changes in patient-reported outcome, pain scores, or wrist and hand function over time.
Despite these limitations, we demonstrate satisfactory outcomes using either radial shortening or VBG in the treatment of adolescent Kienböck's disease based on clinical, radiographic, and patient-reported outcomes. This is the largest case series to date and further expands on the role of revascularization procedures in adolescent patients, of which long-term outcomes are largely unknown.
Funding Statement
Funding None.
Conflict of Interest None declared.
Note
All work was performed at the Mayo Clinic, Rochester, MN.
Ethical Approval
Institutional ethics committee approved the use of personal health data.
All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008.
Informed consent was obtained from all patients included in the study.
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