|Ahead of print publication
Kienbock’s Disease with Volar Intercalated Segment Instability: A Therapeutic Dilemma
Sumit Arora1, Ajeesh Sankaran2, Anil Dhal3
1 Department of Orthopaedic Surgery, Maulana Azad Medical College & Associated Lok Nayak Hospital, New Delhi, India
2 Department of Hand & Reconstructive Microsurgery, KIMS Al Shifa Hospital, Perinthalmanna, Kerala, India
3 Department of Orthopaedics, ESIC Medical College & Hospital, Faridabad, Haryana, India
|Date of Submission||13-Nov-2020|
|Date of Decision||28-Jan-2021|
|Date of Acceptance||29-Jan-2021|
|Date of Web Publication||09-Jun-2021|
Department of Orthopaedic Surgery, Maulana Azad Medical College & Associated Lok Nayak Hospital, New Delhi
Source of Support: None, Conflict of Interest: None
The etiology of avascular necrosis of the lunate (Kienbock disease) remains unclear despite long clinical experience. Very rarely, it may be associated with carpal instability. We describe a case of 45-year-old female who had Kienbock disease with associated volar intercalated segment instability, detailing how the radiographic features can often cause a dilemma in the staging and management of this enigmatic problem.
Keywords: Avascular necrosis, carpal instability, Kienbock disease, lunate, volar intercalated segment instability
|How to cite this URL:|
Arora S, Sankaran A, Dhal A. Kienbock’s Disease with Volar Intercalated Segment Instability: A Therapeutic Dilemma. MAMC J Med Sci [Epub ahead of print] [cited 2021 Jun 13]. Available from: https://www.mamcjms.in/preprintarticle.asp?id=318087
| Introduction|| |
Avascular necrosis of lunate was originally described by Peste in 1843. The condition was named after Roger Kienbock, the Viennese radiologist who first studied the radiographic changes. The etiology of this well known condition has long been studied, but the origin and natural history remains unclear. No consistent correlation with any disease process has been noted, though it has been associated with scleroderma, sickle cell anemia, steroid use, and systemic lupus erythematosus (SLE).
Fragmentation of the lunate leads to loss of mechanical strength of the carpal central column, with a kinematic delinking in the proximal row. The scapholunate and lunotriquetral ligaments may eventually fail, causing scaphoid flexion and proximal triquetral migration. Volar intercalated segment instability (VISI) being more likely if the lunotriquetral ligaments fail. Though the scaphoid hyperflexes this ought not to be confused with a typical Dorsal Intercalated Segment Instability (DISI) finding. The resultant carpal instability (neutral/VISI/DISI) also depends on which part of the lunate (palmar/dorsal/global) is mainly involved.
We present a rare case of Kienbock disease complicated with VISI. In addition, we describe the importance of true lateral view of the wrist in the staging of disease process in such situations. The patient was subsequently treated with a revascularization procedure.
| Case Report|| |
A 45-year-old right hand dominant house wife presented as an outpatient with complaint of pain in left wrist for past 6 months. The pain was insidious in onset, gradually progressive and dull aching in nature. There was no diurnal variation. The pain aggravated with activity and was relieved with rest or oral analgesics. There was a subjective weakness in hand grip. There was no history of wrist trauma. Her routine activity profile also did not suggest repetitive wrist trauma. She had no history of persistent fever, constitutional symptoms of tuberculosis, steroid intake, and any skin lesions suggestive of scleroderma or SLE. Her family history and personal history were noncontributory.
General physical examination was unremarkable. There was presence of swelling over the dorsum of the involved wrist without any apparent deformity. Tenderness could be elicited over the dorsal radiolunate area. She complained of increase in pain on resisted dorsiflexion of wrist with the fingers extended, suggesting radioscapholunate region involvement. Active dorsiflexion was limited to 75° from neutral, while palmar flexion showed more severe restriction with just 30° of available arc from the neutral. Lichtman test for midcarpal instability was negative, as were Scapholunate shear tests and Watson’s scaphoid shift tests. Both the radial and ulnar pulses were normally palpable on both the sides. The motor and sensory examination of the left upper limb was essentially normal.
Posteroanterior and lateral view radiographs of the wrist were obtained. The scaphoid showed a “palmar ring” sign on the Posteroanterior (PA) view, suggesting it to be hyperflexed relative to the axis of the radius. The lunate appeared irregular, deformed, and severely collapsed [Figure 1]A. The capitate had migrated proximally. Ulnar variance was negative bilaterally (−3 mm).
|Figure 1 (A) Posteroanterior radiograph of the left wrist showing irregularity and collapse of lunate with “palmar ring” sign in scaphoid (arrows). (B) Lateral view radiograph of the left wrist showing flexed lunate (arrow), suggestive of VISI carpal instability pattern (a note may be made of the relatively preserved height of the lunate).|
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The lateral view revealed flexion of the scaphoid with a scaphoradial angle of 75° on the left as compared to 60° on the right [Figure 1]B. The radiolunate angle was also increased (35° on the left against 5° on the right). These findings on lateral view were overlooked initially. During preoperative planning, on a closer look at the lateral view, lunate hyperflexion relative to the radius was noticed. The deformation quotient was calculated to be 0.47 (normal = 0.50–0.56). On hindsight, similar findings were noticed on the initial X-rays.
Magnetic Resonance (MR) imaging of the left wrist was performed with gadolinium contrast. The lunate displayed low marrow signal intensity on T1-W, T2-W, and Short Tau Inversion Recovery (STIR) images. It revealed a significant collapse of the left lunate in coronal plane [Figure 2]A, whereas sagittal plane revealed relative preservation of its height along with increased flexion of the lunate and proximal migration of the capitate [Figure 2]B. Thickened enhancing synovium with effusion was also noted in the radiocarpal and carpal joints.
|Figure 2 (A) MRI T1-W coronal image of the left wrist showing hypointense lunate with apparent decreased height (arrow). (B) MRI T1-W sagittal image of the left wrist showing lunate hyperflexion with relative preservation of its height.|
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The patient was initially explained that the success rate of revascularization would be unpredictable and a need for salvage procedure (scaphocapitate fusion or scaphotrapeziotrapezoid fusion) was debated. On reassessment of radiographs and Magnetic Resonance Imaging (MRI) during preoperative planning, it was realized that volar flexion of lunate (VISI) made it to appear collapsed in PA views. She underwent the revascularization procedure by vascularized bone grafting from the dorsal distal radius, based on the fourth extensor compartment artery [Figure 3]. At 6 months follow-up, the patient reported complete pain relief and was able to carry out daily routine activities.
|Figure 3 Intraoperative photograph demonstrating arterial arcade over dorsal wrist with the fourth and fifth extensor compartment arteries.|
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A written, informed consent was obtained from the patient authorizing treatment, photographic documentation, and radiographic examination. She was also informed that data concerning the case would be submitted for publication, and she consented.
| Discussion|| |
Kienbock disease appears to be multifactorial in causation. Gelberman et al. have supported a correlation between ulnar negative variance and Kienbock disease. However, this view has been challenged in the last two decades., Tsuge and Nakamura have suggested that the slope of distal radius may be a potential cause for Kienbock disease and they observed that radial inclination was lower in such patients.
Patients are usually young males with pain and stiffness in the dominant wrist. Tenderness may be present over the radiolunate area with effusion. Radiographs show an increased bone density of the lunate. MRI will demonstrate decreased signal intensity in T1 images, over the entire lunate. The most popular method of staging was initially described by Stahl that was later modified by Lichtman et al. The system uses plain radiographs and MRI findings with utility in deciding the plan of management. In stage I, radiographs are normal but MRI will show decreased T1 signal intensity. Bone scans show increased radionuclide uptake. In stage II, the lunate is sclerosed but not collapsed. Stage III is subdivided into stages A and B. In IIIA, there is collapse of the lunate though the rest of carpals are normal. In IIIB, the capitate migrates proximally while the scaphoid assumes a flexed position. In stage IV, arthritic changes appear in the radiocarpal and midcarpal joints.
In our patient, the proximal migration of capitate was largely because of hyperflexion of lunate rather than collapse of lunate that produced a dilemma in staging of the entity. Therefore, we emphasize the importance of orthogonal views of wrist, especially true lateral views, in such cases.
Carpal instability was initially held to be synonymous with malalignment, meaning that a wrist was considered unstable if the carpal bones showed an alteration in the sagittal or frontal alignment. However, Linscheid et al. suggested that all alterations in carpal alignment are not pathologic. The current concept defines that the wrist is unstable if it is incapable of preserving a normal kinetic or kinematic connection between the radius, carpals, and metacarpals. Therefore, abnormal carpal motion (dyskinetics) also implies instability. Avascular necrosis, inflammatory arthritides, and other processes alter the shape of carpal bones or modify the interaction between the carpals and result in instability. Several patterns of direction of malalignment have been described, the common ones being DISI, VISI, and others like ulnar, radial, or dorsal translocation.
The hyperflexion of the lunate, in our case, seen most prominently on the lateral wrist radiograph could be the result of a differential collapse of the lunate (volar more than dorsal) resulting in a gradual squeezing out of the lunate by the radius proximally and the capitate distally which resulted in an apparent dorsal subluxation. This would only be possible by a concomitant lengthening of the restraining ligaments which may have accrued out of the decreased blood supply of the lunate itself coupled with continuous gradual distraction caused by the squeezing out of the lunate. Since the triquetrum is tightly constrained to the proximal row (vide the triquetro–capito–hamate ligament), failure of the lunotriquetral ligament complex causes the lunate scaphoid unit to fall into flexion. This produces the typical VISI pattern of instability, with a concomitant tendency of capitate volar subluxation.
In the postoperative radiographs as well, the features persisted as shape of lunate and the ligamentous “slack” were not amenable to reconstruction. Nevertheless, we present an unusual case of Kienbock disease complicated with VISI and emphasize the importance of lateral view radiographs of the wrist in the management of such cases. Since collapse of lunate was not significant, our patient was treated with a revascularization procedure.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Peste JL. Discussion. Bull Soc Anat 1843;18:169.
Kienbock R. Uber traumatische Malazie des Monatbeins und ihre Folgezustande: Entartungsformen and Kompressionfrakturen [in German]. Fortschrit Rontgenstrallen 1910;16:77.
Garcia-Elias M. Carpal instability. In Wolffe SW, Hotchkiss RN, Pederson WC, Kozin SH, eds. Green’s Operative Hand Surgery. 6th
ed. Philadelphia: Churchill Livingstone; 2011. p. 465–522.
Gelberman RH, Bauman TD, Menon J. The vascularity of the lunate bone and Kienbock’s disease. J Hand Surg Am 1980;5:272-8.
D’Hoore K, De Smet L, Verellen K, Vral J, Fabry G. Negative ulnar variance is not a risk factor for Kienbock’s disease. J Hand Surg Am 1994;19:229-31.
Nakamura R, Watanabe K, Tsunoda K, Miura T. Radial osteotomy for Kienbock’s disease evaluated by magnetic resonance imaging: 24 cases followed for 1–3 years. Acta Orthop Scand 1993;64:207-11.
Tsuge S, Nakamura R. Anatomical risk factors for Kienbock’s disease. J Hand Surg Br 1993;18:70-5.
Stahl F. On lunatomalacia (Kienbock’s disease): a clinical and roentgenological study, especially on the pathogenesis and the late results of immobilization treatment. Acta Chir Scand 1947;95(Suppl 126):3.
Lichtman DM, Mack GR, MacDonald RI, Gunther SF, Wilson JN. Kienbock’s disease: the role of silicone replacement arthroplasty. J Bone Joint Surg Am. 1977;59:899.
Linscheid RL, Dobyns JH, Beabout JW, Bryan RS. Traumatic instability of the wrist. Diagnosis, classification, and pathomechanics. J Bone Joint Surg Am. 1972;54:1612-32.
Anatomy and Biomechanics Committee of the IFSSH. Position Statement: definition of carpal instability. J Hand Surg Am. 1999;24:866-7.
[Figure 1], [Figure 2], [Figure 3]