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Carpal Kinematics in Madelung Deformity

Published:April 10, 2021DOI:https://doi.org/10.1016/j.jhsa.2020.11.016

      Purpose

      Various skeletal and soft tissue abnormalities have been identified in Madelung deformity and have been hypothesized to play a causal role in its progressive symptomatology; however, our pathological understanding of these changes remains limited. In this study, we biomechanically assessed the Madelung deformity wrist, using 4-dimensional computed tomography imaging.

      Methods

      Nine Madelung deformity wrists (5 patients; age, 24 ± 5 y) and 18 healthy wrists (9 volunteers; age, 28 ± 3 y) underwent 4-dimensional imaging during flexion-extension motion and radioulnar deviation. Carpal kinematics and radiocarpal joint parameters were quantified and compared.

      Results

      In Madelung deformity wrists, significantly decreased rotation was seen in the lunate (–4.6°) and the triquetrum (–4.8°) during flexion-extension motion. During radioulnar deviation, significant decreases were visible in lunate bone translation (–0.7 mm), triquetrum bone translation (–0.6 mm), and triquetrum bone rotation (–1.9°). Patients had significantly decreased articulating surface areas of the scaphoid (1.4 ± 0.2 cm2 versus 1.6 ± 0.2 cm2) and lunate (1.2 ± 0.4 cm2 versus 1.5 ± 0.3 cm2) fossa, and significantly increased radioscaphoid (1.3 ± 0.1 mm versus 1.2 ± 0.1 mm) and radiolunate (1.6 ± 0.2 mm versus 1.3 ± 0.3 mm) joint space thicknesses.

      Conclusions

      There is a decreased mobility of the lunate and triquetrum bones in Madelung deformity.

      Clinical relevance

      Four-dimensional imaging could be used in future studies that investigate the effect of surgical ligament release on carpal kinematics and subsequent wrist mobility.

      Key words

      Madelung deformity is a rare congenital deformity
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      Die spontane Subluxation der Hand nach vorne.
      that commonly manifests bilaterally
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      and emerges clinically in early adolescence.
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      Symptoms include wrist pain and a reduced range of motion (ROM),
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      both important factors in deciding whether a patient could benefit from undergoing surgical treatment.
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      Owing to its rarity, with incidence and prevalence still unknown, our understanding of Madelung deformity is incomplete.
      • Flatt A.E.
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      There is no consensus among clinicians and researchers with regard to various clinical aspects such as etiology,
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      Previous studies have established a spectrum of skeletal abnormalities, particularly in the distal radius, which have been quantified for use in diagnosis.
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      Quantitative three-dimensional assessment of Madelung deformity.
      Furthermore, imaging studies have revealed various soft tissue abnormalities, including the volar radiolunate ligament known as Vickers ligament and the anomalous volar radiotriquetral ligament.
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      • Vieth V.
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      • Zlotolow D.A.
      Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics.
      These anatomical changes are thought to play a causal role in the progressive symptomatology of the condition, with the purpose of surgery being to restore the anatomical configuration and normalize the anatomy in order to improve wrist biomechanics.
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      Whereas previous biomechanical studies have substantially expanded our knowledge of wrist anatomy,
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      Carpal kinematics after proximal row carpectomy.
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      • Streekstra G.J.
      Dynamic in vivo evaluation of radiocarpal contact after a 4-corner arthrodesis.
      no in vitro or in vivo biomechanical studies have been performed for Madelung deformity.
      The objective of this study was to investigate the wrist biomechanics of Madelung deformity patients, using 4-dimensional computed tomography (CT) imaging during flexion-extension motion and radioulnar deviation, and comparing those biomechanical features with wrists of healthy volunteers.

      Methods

      Setting and study population

      For this study, we retrospectively included patients with Madelung deformity, for whom 4-dimensional CT scans of 1 or both wrists were available. All Madelung deformity patients visiting our clinic undergo 4-dimensional CT imaging to visualize carpal bone kinematics based on a previously developed protocol, independent of the severity of the deformity.
      • Dobbe J.G.G.
      • de Roo M.G.A.
      • Visschers J.C.
      • Strackee S.D.
      • Streekstra G.J.
      Evaluation of a quantitative method for carpal motion analysis using clinical 3-D and 4-D CT protocols.
      In the diagnostic work-up, the McCarroll et al criteria
      • McCarroll Jr., H.R.
      • James M.A.
      • Newmeyer III, W.L.
      • Molitor F.
      • Manske P.R.
      Madelung's deformity: quantitative assessment of x-ray deformity.
      were used, quantifying the deformity with 4 measurements: ulnar tilt, lunate subsidence, lunate fossa angle, and palmar carpal displacement. Ali et al
      • Ali S.
      • Kaplan S.
      • Kaufman T.
      • Fenerty S.
      • Kozin S.
      • Zlotolow D.A.
      Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics.
      have previously hypothesized the deformity to be true Madelung deformity in the presence of a Vickers ligament or if a patient is bilaterally affected with an underlying diagnosis of Leri-Weill dyschondrosteosis,
      • Belin V.
      • Cusin V.
      • Viot G.
      • et al.
      SHOX mutations in dyschondrosteosis (Leri-Weill syndrome).
      although there is no consensus on this definition. Patient charts were reviewed to determine any underlying genetic disorders and surgical notes were reviewed to assess the presence of Vickers ligament. For patients who did not undergo surgical treatment, we reviewed preoperative x-ray and CT imaging instead to identify the ligamentous origin.
      • Ali S.
      • Kaplan S.
      • Kaufman T.
      • Fenerty S.
      • Kozin S.
      • Zlotolow D.A.
      Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics.
      We excluded patients with a posttraumatic Madelung deformity and wrists that had previously undergone surgical correction. A total of 5 patients were eligible for inclusion; all with bilateral Madelung deformity. After excluding 1 wrist (previous corrective osteotomy of the radius), this resulted in a total of 9 included wrists. In addition, previously acquired 4-dimensional CT scans of 9 healthy female volunteers (18 wrists) were included in the kinematic analysis for comparison. None of the healthy volunteers had a medical history of skeletal disorders or a surgical history of wrist interventions. Volunteers with hypermobility, assessed using the Beighton score,
      • Smits-Engelsman B.
      • Klerks M.
      • Kirby A.
      Beighton score: a valid measure for generalized hypermobility in children.
      were excluded. This study was approved by our medical center institutional review board.

      Image acquisition

      The 4-dimensional CT scans were acquired using a Philips Brilliance 64 CT scanner (Philips, Cleveland, OH). First, a static CT scan was performed of the wrist in neutral position (120 kV, 75 mAs). Next, the participant gripped the handle of a custom-made positioning device (Fig. 1), limiting wrist movement to either flexion-extension or radioulnar deviation. Participants performed each motion over 12 seconds while dynamic 4-dimensional CT scans were acquired (120 kV, 30 mAs; collimation, 64 × 0.625 mm; axial field of view, 4 cm; rotation time, 0.4 s), resulting in a total of 30 reconstructions per motion. The static high-quality CT scan was used for segmentation; the dynamic low-quality CT scans were used for position detection. Participants received a total dose of 0.3 mSv.
      Figure thumbnail gr1
      Figure 1Wrist positioning device to keep the carpus in the field of view during 4-dimensional imaging. The patient can move the wrist about either the flexion-extension axis or the radioulnar deviation axis.

      Image segmentation and position detection

      The static CT scan of the wrist in neutral position was segmented using a custom-made software package
      • Dobbe J.G.G.
      • de Roo M.G.A.
      • Visschers J.C.
      • Strackee S.D.
      • Streekstra G.J.
      Evaluation of a quantitative method for carpal motion analysis using clinical 3-D and 4-D CT protocols.
      to obtain virtual 3-dimensional models of the radius, ulna, scaphoid, lunate, triquetrum, and capitate bones. The scaphoid, lunate, and triquetrum were used to assess radiocarpal motion; the capitate was used to assess overall wrist motion. The virtual bone models were aligned with the dynamic 4-dimensional CT scans (30 per motion) to quantify the position of each carpal bone during movement using 2 parameters: translation (x, y, and z coordinates of the virtual bone models’ centroid), and rotation (φ, θ, and ψ angles relative to the neutral position). Total translation was calculated using the formula x2+y2+z2 and total rotation was calculated using the formula φ2+θ2+ψ2.

      Image processing and computation

      The virtual 3-dimensional models and corresponding kinematics parameters were exported and further processed using software programmed in MATLAB R2018b (MathWorks Inc., Natick, MA). This software performed the following functions automatically: wrist alignment, detection of radial and ulnar landmarks (eg, radial styloid process, ulnar styloid process), visualization of the wrist during motion (Fig. 2), and the computations described later.
      Figure thumbnail gr2
      Figure 2Visualizing movement of virtually segmented wrist models. Colors indicate different positions during motion (blue, first position; orange, last position).
      To quantify the position of the wrist during motion in flexion-extension or radioulnar deviation angles, we first determined the longitudinal axis of the capitate bone in neutral position. Next, we calculated the angle in degrees between this neutral axis and the dynamic longitudinal axis of the capitate bone during each of the 30 positions per motion. To visualize a full motion in a graph, flexion and radial deviation angles were represented as negative values; extension and ulnar deviation angles were represented as positive values.
      Articular surface area and joint space thickness (Fig. 3) were calculated using a previously described method.
      • Peymani A.
      • Foumani M.
      • Dobbe J.G.G.
      • Strackee S.D.
      • Streekstra G.J.
      Four-dimensional rotational radiographic scanning of the wrist in patients after proximal row carpectomy.
      ,
      • Foumani M.
      • Strackee S.D.
      • van de Giessen M.
      • Jonges R.
      • Blankevoort L.
      • Streekstra G.J.
      In-vivo dynamic and static three-dimensional joint space distance maps for assessment of cartilage thickness in the radiocarpal joint.
      Briefly, for each of the 30 positions during a motion, we calculate for each point on the radius the nearest point to the scaphoid or lunate. Next, these points are filtered using 2 pragmatically chosen constraints: (1) a maximum distance of 4 mm between opposing points; (2) a maximum angle difference of 15° between the normal lines of opposing points. The articular surface area per motion is acquired by merging the points of 30 positions; a combined version is calculated by merging the points of all 60 positions from 2 motions (flexion-extension and radio-ulnar deviation). For each position within the motion trajectory, the minimum distance to the opposing point is taken for each point in the articular surface area. The mean of these minimum distances provides the joint space thickness, defined as the articular cartilage thickness between the radius and the scaphoid (radioscaphoid joint space thickness) or between the radius and the lunate (radiolunate joint space thickness).
      Figure thumbnail gr3
      Figure 3Articular surface area and joint space thickness.
      Carpal bone kinematics, articular surface area, and joint space thickness are visualized in Video E1 and Figures 4 and 5.
      Figure thumbnail gr4
      Figure 4Carpal bone kinematics during flexion-extension motion.
      Figure thumbnail gr5
      Figure 5Carpal bone kinematics during radioulnar deviation.

      Data processing and statistical analysis

      Python v3.7.2 (Python Software Foundation. Python Language Reference, version 3.7.2) was used to process MATLAB data files and to perform statistical analyses and graphical data visualization using the SciPy, StatsModels, and Seaborn packages.
      For ROM, articular surface area, and joint space thickness, we compared the means of the calculated parameters between Madelung deformity wrists and healthy wrists. Data normality was determined using the Shapiro-Wilk test. If a variable was normally distributed in both groups, an independent samples t test (variances equal) or Welch t test (variances not equal) was performed. Equality of variances was assessed using the Levene test. If a variable was not normally distributed, the Mann-Whitney U test was performed.
      To compare carpal bone kinematics while adjusting for ROM (flexion-extension and radioulnar deviation angles), we developed mixed-effects models for each combination of 3 proximal carpal bones (scaphoid, lunate, and triquetrum), 2 distinct motions (flexion-extension and radioulnar deviation), and 2 outcomes of interest (total translation and total rotation); this resulted in a total of 12 models. As fixed-effects, we entered angle (ROM) and wrist type (healthy or Madelung). In addition, to account for possible size differences between Madelung patients and healthy volunteers, we added wrist size as a fixed effect, defined as the absolute distance between the centroids of capitate and the lunate. Lastly, to optimally fit each model, we added angle2 and angle3 as fixed effects, depending on the outcome curve being quadratic- or cubic-shaped, respectively. Intercepts for different wrists were added as random effects. The P values were obtained by likelihood ratio tests of the full model including wrist type and the model without wrist type. To determine which specific parameters were responsible for an altered total translation or rotation, we performed separate subanalyses for translation over x, y, and z coordinates (Table E1), and for rotation over φ, θ, and ψ angles (Table E2).

      Results

      Patients with Madelung deformity (n = 5) had a mean age of 23.7 ± 4.9 years; healthy volunteers (n = 9) had a mean age of 28.0 ± 2.6 years. All participants were women. Study characteristics are described in Table 1. Wrists of Madelung deformity patients had a significantly lower maximum angle of flexion (47.9° ± 16.5° versus 72.8° ± 7.2°; P < .05) and radial deviation (12.5° ± 8.5° versus 23.3° ± 5.5°; P < .05) compared with wrists of healthy volunteers (Table 2). No significant differences were found for maximum extension and maximum ulnar deviation.
      Table 1Characteristics of the Study Group
      VariablesMadelung Deformity Patients (n = 5)Healthy Volunteers (n = 9)
      Wrists918
      Age, y (SD)23.7 (4.9)28.0 (2.6)
      Female59
      Bilateral deformity5NA
      Confirmed genetic cause3NA
      Radiographic measurements
       Ulnar tilt, ° (SD)41.1 (19.3)NA
       Lunate subsidence, mm (SD)9.2 (4.2)NA
       Lunate fossa angle, ° (SD)56.5 (17.4)NA
       Palmar carpal displacement, mm (SD)15.5 (7.0)NA
      NA, not applicable.
      Table 2Maximum Range of Motion With Respect to the Capitate Bone
      Madelung Deformity Wrists (n=9)Healthy Wrists (n=18)P Value
      Statistically significant differences (P < .05) shown in bold.
      Flexion, ° (SD)47.9 (16.5)72.8 (7.2)<.05
      Extension, ° (SD)43.8 (12.4)44.4 (5.3).894
      Radial deviation, ° (SD)12.5 (8.5)23.2 (5.5)<.05
      Ulnar deviation, ° (SD)19.2 (7.0)19.8 (10.4).882
      Statistically significant differences (P < .05) shown in bold.
      During flexion-extension motion (Table 3), a significantly decreased rotation was seen in both the lunate bone (difference, –4.6°; 95% confidence interval [95% CI], –7.1 to –2.2; P < .05) and the triquetrum bone (difference, –4.8°; 95% CI, –6.7 to –3.0; P < .05) for Madelung deformity wrists. Carpal kinematics during radioulnar deviation (Table 4) showed significant decreases in lunate bone translation (difference, –0.7 mm; 95% CI, –1.0 to –0.4; P < .05), >triquetrum bone translation (difference, –0.6 mm; 95% CI, –0.9 to –0.3; P < .05), and triquetrum bone rotation (difference, –1.9; 95% CI, –3.7 to –0.1; P < .05). There were no significant differences found for scaphoid bone translation and rotation during flexion-extension motion or radioulnar deviation.
      Table 3Carpal Kinematics During Flexion-Extension Motion
      Mean DifferenceStandard Error95% CIP Value
      Statistically significant differences (P < .05) shown in bold.
      Scaphoid translation, mm–0.30.1–0.5 to 0.0.064
      Scaphoid rotation, °–1.41.0–3.3 to 0.6.182
      Lunate translation, mm–0.20.1–0.4 to 0.0.119
      Lunate rotation, °–4.61.3–7.1 to –2.2<.05
      Triquetrum translation, mm–0.20.2–0.6 to 0.1.248
      Triquetrum rotation, °–4.80.9–6.7 to –3.0<.05
      Statistically significant differences (P < .05) shown in bold.
      Table 4Carpal Kinematics During Radioulnar Deviation
      Mean DifferenceStandard Error95% CIP Value
      Statistically significant differences (P < .05) shown in bold.
      Scaphoid translation, mm–0.30.2–0.7 to 0.1.136
      Scaphoid rotation, degrees2.51.6–0.7 to 5.6.131
      Lunate translation, mm–0.70.2–1.0 to –0.4<.05
      Lunate rotation, degrees–1.41.2–3.9 to 1.0.256
      Triquetrum translation, mm–0.60.2–0.9 to –0.3<.05
      Triquetrum rotation, degrees–1.90.9–3.7 to –0.1<.05
      Statistically significant differences (P < .05) shown in bold.
      Table 5 describes the articular surface area and joint space thickness of the distal radius during motion. Patients with Madelung deformity had significantly decreased articulating surface areas of the scaphoid (1.4 ± 0.2 cm2 versus 1.6 ± 0.2 cm2; P < .05) and lunate fossa (1.2 ± 0.4 cm2 versus 1.5 ± 0.3 cm2; P<0.05), and significantly increased radioscaphoid (1.3 ± 0.1 mm versus 1.2 ± 0. mm; P < .05) and radiolunate joint space thickness (1.6 ± 0.2 mm versus 1.3 ± 0.3 mm; P < .05).
      Table 5Articular Surface Area and Joint Space Thickness
      Madelung Deformity Wrists (n = 9)Healthy Wrists (n = 18)P Value
      Statistically significant differences (P < .05) shown in bold.
      Articular surface area, cm2 (SD)
      Scaphoid fossa
       Combined
      Combined for 2 motions.
      1.4 (0.2)1.6 (0.2)<.05
       Flexion-extension motion1.3 (0.2)1.5 (0.2)<.05
       Radioulnar deviation1.3 (0.1)1.5 (0.1)<.05
      Lunate fossa
       Combined
      Combined for 2 motions.
      1.2 (0.4)1.5 (0.3)<.05
       Flexion-extension motion1.1 (0.4)1.4 (0.3).097
       Radioulnar deviation1.2 (0.4)1.5 (0.3)<.05
      Joint space thickness, mm (SD)
      Radioscaphoid joint
       Combined
      Combined for 2 motions.
      1.3 (0.1)1.2 (0.1)<.05
       Flexion-extension motion1.4 (0.1)1.2 (0.1)<.05
       Radioulnar deviation1.4 (0.1)1.4 (0.1).643
      Radiolunate joint
       Combined
      Combined for 2 motions.
      1.6 (0.2)1.3 (0.3)<.05
       Flexion-extension motion1.7 (0.2)1.4 (0.2)<.05
       Radioulnar deviation1.7 (0.3)1.4 (0.3)<.05
      Statistically significant differences (P < .05) shown in bold.
      Combined for 2 motions.

      Discussion

      In this study, we imaged patients’ wrist bones during flexion-extension motion and radioulnar deviation through a previously developed 4-dimensional CT protocol. In comparison with wrists of healthy volunteers, wrists of Madelung deformity patients have decreased mobility of the lunate and triquetrum bones. The articular surface areas of the scaphoid and lunate fossa are decreased, and radiocarpal cartilage thickness does not significantly differ from healthy wrists.
      A major strength of this in vivo study is that all measurements were performed automatically through self-developed software algorithms
      • Peymani A.
      • Foumani M.
      • Dobbe J.G.G.
      • Strackee S.D.
      • Streekstra G.J.
      Four-dimensional rotational radiographic scanning of the wrist in patients after proximal row carpectomy.
      leading to an objective assessment in a dynamic setup. Another strength is that the mixed-model analysis of 4-dimensional CT data allows for the quantification of individual carpal bone mobility, while considering the differences in wrist ROM between patients and volunteers. Because wrist ROM is intrinsically linked to carpal bone mobility, correcting for this parameter in our analyses shows that the reported differences in mobility occur despite, and not because of, differences in ROM. A limitation of this study is the small number of wrists (n = 9) and corresponding limited statistical power because most of the patients in our database could not be included owing to having previously undergone surgical procedures to correct the deformity. Another limitation was that 2 patients received growth hormone therapy, shown to be effective in the treatment of short stature associated with SHOX gene deficiency,
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      Effectiveness of the combined recombinant human growth hormone and gonadotropin-releasing hormone analog therapy in pubertal patients with short stature due to SHOX deficiency.
      possibly influencing wrist kinematics owing to altered skeletal growth. Lastly, the investigative scope of this study was limited to flexion-extension motion and radioulnar deviation (radiocarpal articulation), yet multiple studies have shown decreased mobility to be particularly prominent in forearm pronation and supination (radioulnar articulation).
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      The configuration of our 4-dimensional CT protocol did not allow for capturing kinematics during forearm rotation because of motion artifacts.
      Our findings in regards to the decreased radial deviation seen in patients seem to be in concordance with previous findings.
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      Furthermore, we found patients to have lower maximum angles of flexion with no significant differences for extension. Previous studies that performed preoperative goniometer measurements in a clinical setting found decreases to be primarily in wrist extension rather than flexion,
      • Ranawat C.S.
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      • Straub L.R.
      Madelung's deformity. An end-result study of surgical treatment.
      with extension ranging from 32° to 49°.
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      Madelung's deformity: a spectrum of presentation.
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      Radial opening wedge osteotomy in Madelung's deformity.
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      • Laredo Filho Jr., J.
      Osteotomy of the radius and ulna for the Madelung deformity.
      ,
      • Bruno R.J.
      • Blank J.E.
      • Ruby L.K.
      • Cassidy C.
      • Cohen G.
      • Bergfield T.G.
      Treatment of Madelung's deformity in adults by ulna reduction osteotomy.
      This discrepancy is likely a result of our automatic 3-dimensional measurements, in which our 0-point is defined by the orientation of the capitate with the wrist in a neutral position. One of the clinically visible features of Madelung deformity is the palmar displacement of the carpus.
      • Henry A.
      • Thorburn M.J.
      Madelung's deformity. A clinical and cytogenetic study.
      ,
      • McCarroll Jr., H.R.
      • James M.A.
      • Newmeyer III, W.L.
      • Manske P.R.
      Madelung's deformity: diagnostic thresholds of radiographic measurements.
      This is also visible in the configuration of the virtual bones in 3-dimensional space, in which the capitate is palmarly rotated in a neutral wrist position. Therefore, this shift of the 0-point toward flexion translates into relatively higher degrees of extension and, thus, relatively lower degrees of flexion in our 3-dimensional measurements.
      Clinically measured decreases of ROM have been extensively quantified,
      • Ranawat C.S.
      • DeFiore J.
      • Straub L.R.
      Madelung's deformity. An end-result study of surgical treatment.
      • Salon A.
      • Serra M.
      • Pouliquen J.C.
      Long-term follow-up of surgical correction of Madelung's deformity with conservation of the distal radioulnar joint in teenagers.
      • Zebala L.P.
      • Manske P.R.
      • Goldfarb C.A.
      Madelung's deformity: a spectrum of presentation.
      • Murphy M.S.
      • Linscheid R.L.
      • Dobyns J.H.
      • Peterson H.A.
      Radial opening wedge osteotomy in Madelung's deformity.
      • dos Reis F.B.
      • Katchburian M.V.
      • Faloppa F.
      • Albertoni W.M.
      • Laredo Filho Jr., J.
      Osteotomy of the radius and ulna for the Madelung deformity.
      ,
      • Bruno R.J.
      • Blank J.E.
      • Ruby L.K.
      • Cassidy C.
      • Cohen G.
      • Bergfield T.G.
      Treatment of Madelung's deformity in adults by ulna reduction osteotomy.
      but it is still unknown whether these are caused by pain, intrinsic mechanical properties, or other reasons. In our study, the lunate and triquetrum show decreased mobility during flexion-extension motion and radioulnar deviation, yet scaphoid bone mobility remains normal. We hypothesize that scaphoid and lunate kinematics should be closely intertwined owing to the ligamentous connections of the scapholunate joint.
      • Kitay A.
      • Wolfe S.W.
      Scapholunate instability: current concepts in diagnosis and management.
      However, it has been demonstrated in vivo that these interosseous ligaments allow for the occurrence of considerable multiplanar motion between the 2 bones,
      • Wolfe S.W.
      • Neu C.
      • Crisco J.J.
      In vivo scaphoid, lunate, and capitate kinematics in flexion and in extension.
      resulting in comparable, yet distinct, motion patterns. Interestingly, the lunate and the triquetrum are the only carpal bones for which anomalous ligaments have been identified.
      • Cook P.A.
      • Yu J.S.
      • Wiand W.
      • et al.
      Madelung deformity in skeletally immature patients: morphologic assessment using radiography, CT, and MRI.
      ,
      • Vickers D.
      • Nielsen G.
      Madelung deformity: surgical prophylaxis (physiolysis) during the late growth period by resection of the dyschondrosteosis lesion.
      • Stehling C.
      • Langer M.
      • Nassenstein I.
      • Bachmann R.
      • Heindel W.
      • Vieth V.
      High resolution 3.0 Tesla MR imaging findings in patients with bilateral Madelung's deformity.
      • Ali S.
      • Kaplan S.
      • Kaufman T.
      • Fenerty S.
      • Kozin S.
      • Zlotolow D.A.
      Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics.
      ,
      • Hanson T.J.
      • Murthy N.S.
      • Shin A.Y.
      • Kakar S.
      • Collins M.S.
      MRI appearance of the anomalous volar radiotriquetral ligament in true Madelung deformity.
      In addition to the established skeletal abnormalities, Vickers et al
      • Vickers D.
      • Nielsen G.
      Madelung deformity: surgical prophylaxis (physiolysis) during the late growth period by resection of the dyschondrosteosis lesion.
      were the first to report on a thick volar ligament that firmly restrains the lunate to the radius. Cook et al
      • Cook P.A.
      • Yu J.S.
      • Wiand W.
      • et al.
      Madelung deformity in skeletally immature patients: morphologic assessment using radiography, CT, and MRI.
      identified the radiotriquetral ligament, extending from the radius to the volar aspect of the triquetrum. Although their occurrence has not been reported consistently,
      • Peymani A.
      • Johnson A.R.
      • Dowlatshahi A.S.
      • et al.
      Surgical management of Madelung deformity: a systematic review.
      imaging studies have confirmed their presence in true bilateral Madelung deformity, and their absence in acquired Madelung-type or pseudo–Madelung deformity.
      • Stehling C.
      • Langer M.
      • Nassenstein I.
      • Bachmann R.
      • Heindel W.
      • Vieth V.
      High resolution 3.0 Tesla MR imaging findings in patients with bilateral Madelung's deformity.
      ,
      • Ali S.
      • Kaplan S.
      • Kaufman T.
      • Fenerty S.
      • Kozin S.
      • Zlotolow D.A.
      Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics.
      ,
      • Hanson T.J.
      • Murthy N.S.
      • Shin A.Y.
      • Kakar S.
      • Collins M.S.
      MRI appearance of the anomalous volar radiotriquetral ligament in true Madelung deformity.
      Ali et al
      • Ali S.
      • Kaplan S.
      • Kaufman T.
      • Fenerty S.
      • Kozin S.
      • Zlotolow D.A.
      Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics.
      have previously hypothesized the deformity to be true in the presence of a Vickers ligament or if a patient is bilaterally affected with an underlying diagnosis of Leri-Weill dyschondrosteosis.
      • Belin V.
      • Cusin V.
      • Viot G.
      • et al.
      SHOX mutations in dyschondrosteosis (Leri-Weill syndrome).
      We confirmed ligamentous anomalies in 1 patient during surgery. Three other patients exhibited the characteristic radiolucent flame-shaped notch on imaging, revealed to represent the ligament origin.
      • Kozin S.H.
      • Zlotolow D.A.
      Madelung deformity.
      ,
      • Créteur V.
      • Madani A.
      • Bianchi S.
      Sonographic findings in adult congenital madelung deformity: a case study.
      ,
      • Babu S.
      • Turner J.
      • Seewoonarain S.
      • Chougule S.
      Madelung's deformity of the wrist—current concepts and future directions.
      However, we did not assess each patient using appropriate imaging techniques such as magnetic resonance imaging, which is better suited to confirm ligamentous anomalies.
      • Cook P.A.
      • Yu J.S.
      • Wiand W.
      • et al.
      Madelung deformity in skeletally immature patients: morphologic assessment using radiography, CT, and MRI.
      ,
      • Vickers D.
      • Nielsen G.
      Madelung deformity: surgical prophylaxis (physiolysis) during the late growth period by resection of the dyschondrosteosis lesion.
      ,
      • Hanson T.J.
      • Murthy N.S.
      • Shin A.Y.
      • Kakar S.
      • Collins M.S.
      MRI appearance of the anomalous volar radiotriquetral ligament in true Madelung deformity.
      Whereas retrospectively searching for this abnormality could hypothetically bias our objectivity, the ligament origin is quite distinguishable from any normal anatomical variations.
      • Ali S.
      • Kaplan S.
      • Kaufman T.
      • Fenerty S.
      • Kozin S.
      • Zlotolow D.A.
      Madelung deformity and Madelung-type deformities: a review of the clinical and radiological characteristics.
      Studies have reported an increased supination of 17° to 23° after surgical release, but strong evidence is lacking.
      • Vickers D.
      • Nielsen G.
      Madelung deformity: surgical prophylaxis (physiolysis) during the late growth period by resection of the dyschondrosteosis lesion.
      ,
      • Otte J.E.
      • Popp J.E.
      • Samora J.B.
      Treatment of Madelung deformity with Vicker ligament release and radial physiolyses: a case series.
      Furthermore, the causative mechanism could be the abnormal skeletal configuration instead of the anomalous ligamentous bands. Because the spectrum of skeletal abnormalities has been shown to vary widely, this could limit the generalizability of conclusions regarding carpal kinematics in a subset of patients.
      • McCarroll H.R.
      • James M.A.
      • Newmeyer III, W.L.
      • Manske P.R.
      Madelung's deformity: quantitative radiographic comparison with normal wrists.
      Prospective studies could reveal the effects of different skeletal configurations by assessing the associations between kinematics and parameters that quantify the skeletal deformity, such as ulnar tilt and lunate fossa angle.
      • McCarroll Jr., H.R.
      • James M.A.
      • Newmeyer III, W.L.
      • Molitor F.
      • Manske P.R.
      Madelung's deformity: quantitative assessment of x-ray deformity.
      The impact of Madelung deformity on the radiocarpal joints has not been previously investigated. Previous studies reported a decreased radiolunate surface area in Madelung deformity patients by quantifying the percentage of the lunate that is in contact with the articular radial surface on static x-ray imaging
      • Murphy M.S.
      • Linscheid R.L.
      • Dobyns J.H.
      • Peterson H.A.
      Radial opening wedge osteotomy in Madelung's deformity.
      or by calculating joint parameters in 3-dimensional space.
      • Peymani A.
      • Dobbe J.G.G.
      • Streekstra G.J.
      • McCarroll H.R.
      • Strackee S.D.
      Quantitative three-dimensional assessment of Madelung deformity.
      In addition to quantifying carpal kinematics, 4-dimensional CT imaging allows a dynamic determination of the articular surface areas and cartilage thickness of the radioscaphoid and radiolunate joints.
      • Foumani M.
      • Strackee S.D.
      • van de Giessen M.
      • Jonges R.
      • Blankevoort L.
      • Streekstra G.J.
      In-vivo dynamic and static three-dimensional joint space distance maps for assessment of cartilage thickness in the radiocarpal joint.
      Both joints showed significantly smaller surface areas in patients, especially the lunate fossa. Given the decreased ROM during flexion-extension and radioulnar deviation, it is expected that a smaller contact area is being utilized. Also, it has been hypothesized that a subset of patients experience pain due to osteoarthritis.
      • Henry A.
      • Thorburn M.J.
      Madelung's deformity. A clinical and cytogenetic study.
      ,
      • Ghatan A.C.
      • Hanel D.P.
      Madelung deformity.
      ,
      • Glard Y.
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      Isolated wedge osteotomy of the ulna for mild Madelung's deformity.
      An abnormal radiocarpal joint could result in altered forces being applied on the wrist that could lead to degeneration of the joints.
      • Weiss K.E.
      • Rodner C.M.
      Osteoarthritis of the wrist.
      Despite the abnormal carpal kinematics seen in this study, radiocarpal cartilage remained intact; our patient group was relatively young (mean age, 23.7 ± 4.9 years) and wrist joint osteoarthritis in the general population has been seen mainly in older individuals.
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      ,
      • Dodge H.J.
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      Age-sex specific prevalence of radiographic abnormalities of the joints of the hands, wrists and cervical spine of adult residents of the Tecumseh, Michigan, Community Health Study area, 1962–1965.
      However, a more plausible explanation could be that, because the deformity slowly progresses until becoming clinically apparent in early adolescence, the wrist can fully utilize its adaptive capacity,
      • Ghatan A.C.
      • Hanel D.P.
      Madelung deformity.
      which has been shown to occur even after major anatomical changes.
      • Peymani A.
      • Foumani M.
      • Dobbe J.G.G.
      • Strackee S.D.
      • Streekstra G.J.
      Four-dimensional rotational radiographic scanning of the wrist in patients after proximal row carpectomy.
      These parameters should be investigated in a larger postsurgical patient group with long-term follow-up.
      In conclusion, radiocarpal kinematics in Madelung deformity are abnormal, showing a decreased mobility of the lunate and triquetrum bones. It remains unknown whether this is caused by the anomalous radiolunate and radiotriquetral ligaments or by the distorted skeletal configuration. Prospective studies could use the 4-dimensional CT analysis to investigate the biomechanical effects of surgical ligament release.

      Supplementary Data

      Appendix A

      Table E1Carpal Kinematics During Flexion-Extension Motion
      Mean DifferenceStandard Error95% CIP Value
      Statistically significant differences (P < .05) shown in bold.
      Scaphoid translation, mm–0.30.1–0.5 to 0.0.064
       X axis00.1–0.2 to 0.1.644
       Y axis0.40.20.0 to 0.7.045
       Z axis0.20.2–0.2 to 0.6.302
      Scaphoid rotation, °–1.41–3.3 to 0.6.182
       X axis–4.51.3–7.1 to –1.9>.999
       Y axis0.20.7–1.2 to 1.6.765
       Z axis–1.80.9–3.6 to 0.0.057
      Lunate translation, mm–0.20.1–0.4 to 0.0.119
       X axis–0.20.1–0.4 to -0.1.006
       Y axis0.00.1–0.2 to 0.2.921
       Z axis0.00.1–0.2 to 0.3.740
      Lunate rotation, °–4.61.3–7.1 to –2.2.001
       X axis–2.81.2–5.2 to –0.5>.999
       Y axis0.50.5–0.4 to 1.5.279
       Z axis–0.30.5–1.3 to 0.6.469
      Triquetrum translation, mm–0.20.2–0.6 to 0.1.248
       X axis–0.20.1–0.4 to –0.1.006
       Y axis0.20.2–0.1 to 0.5.293
       Z axis0.10.1–0.1 to 0.4.348
      Triquetrum rotation, °–4.80.9–6.7 to –3.0<.001
       X axis–2.41.1–4.6 to –0.3.035
       Y axis0.60.6–0.5 to 1.7.271
       Z axis-0.30.6–1.5 to 0.9.607
      Statistically significant differences (P < .05) shown in bold.
      Table E2Carpal Kinematics During Radioulnar Deviation
      Mean DifferenceStandard Error95% CIP Value
      Significant differences shown in bold.
      Scaphoid translation, mm–0.30.2–0.7 to 0.1.136
       X axis0.10.1–0.1 to 0.2.468
       Y axis0.00.1–0.2 to 0.2.876
       Z axis–0.40.2–0.7 to 0.0.058
      Scaphoid rotation, °2.51.6–0.7 to 5.6.131
       X axis–5.41.7–8.7 to –2.1.003
       Y axis3.11.11.0 to 5.2.006
       Z axis–3.21.0–5.2 to –1.3.003
      Lunate translation, mm–0.70.2–1.0 to –0.4<.001
       X axis–0.50.1–0.8 to –0.2.002
       Y axis0.00.1–0.1 to 0.1.771
       Z axis–0.20.1–0.4 to 0.0.108
      Lunate rotation, °–1.41.2–3.9 to 1.0.256
       X axis–2.61.2–5.0 to –0.2.041
       Y axis3.30.91.5 to 5.0.001
       Z axis–2.70.7–4.1 to –1.3>.999
      Triquetrum translation, mm–0.60.2–0.9 to –0.3.001
       X axis–0.50.2–0.8 to –0.2.002
       Y axis0.60.20.2 to 0.9.002
       Z axis0.20.2–0.2 to 0.6.325
      Triquetrum rotation, °–1.90.9–3.7 to –0.1.049
       X axis–3.31.1–5.4 to –1.1.006
       Y axis2.60.81.0 to 4.2.003
       Z axis–1.40.5–2.4 to –0.4.009
      Significant differences shown in bold.

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