Distraction Histiogenesis for the Treatment of Lichtman Stage II Kienböck s Disease: A Case Report

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1 Tungs Medical Journal 11 (2017) Case Report Distraction Histiogenesis for the Treatment of Lichtman Stage II Kienböck s Disease: A Case Report Wei Cheng Lai, Sheng Chieh Lin, Hung Kai Lo* Department of Orthopaedic Surgery, Chung-Shan Medical University Hospital, Institute of Medicine, Chung-Shan Medical University, Taichung, Taiwan Received: May. 09, 2016; Accepted: Aug. 02, 2016 Abstract Kienböck s disease is a rare disorder of wrist which eventually shows osteonecrosis of the carpal lunate. Untreated, the disease usually results in fragmentation of the lunate, collapse with shortening of the carpus. However, there is no consensus regarding treatment, whether conservatively or surgically. Various surgical treatments have been described with varying outcome. Different stages of the disease also warrant different surgical treatment. Here we report our experience in the treatment of a patient with Lichtman stage II Kienböck s disease. Debridement of necrotic lunate bone was performed, followed by distraction with an external fixation device. Our result indicate favorable functional outcome with improvement in wrist strength and mobility. Further studies are necessary to understand the full potential of our treatment. Key words: Kienböck s disease, distraction histiogenesis, osteonecrosis, lunatomalacia Introduction Kienböck s disease is a disorder of the lunate which occurs mainly among patients between 20 and 40. Various methods of treatment have been used since its description by Robert Kienböck in Patients complained of pain, loss of mobility, and prominence in the area of the wrist. Radiographic evaluation may show isolated changes in the proximal aspect of the lunate, with eventual collapse and fragmentation of the bone. Various surgical procedures have been proposed, depending on the stage of pathological development and on anatomic variables such as ulnar variance and radial inclination [1]. Proposed procedures include radial shortening [2], ulnar lengthening [3], conventional or pedicled vascular bone * Correspondence to: Hung Kai Lo, MD, Department of Orthopaedic Surgery, Chung-Shan Medical University Hospital, Institute of Medicine, Chung-Shan Medical University, Taichung, Taiwan No. 110, Sec. 1, Chien-Kuo N. Road, 402 Taichung, Taiwan (R.O.C.) graft [4], arthrodesis [5], proximal row carpectomy [6] and arthroplasty [7]. In the hope to simplify a surgical procedure that is easily accessible and familiar to general orthopedic surgeons, we applied the principle of distraction histiogenesis as used in Perthes disease in children [8, 9]. We hypothesized that debridement of necrotic bone will start the healing process, which when combined with distraction of the carpus will unload and relief the injured bone, increase vascularity of the region and help maintain the health of the lunate bone. In addition, lunate height is maintained while healing occurs. Case report A 32-year-old Asian man, who is a labor intensive worker, came in with the complaint of left wrist pain, decreased grip strength and limited range of motion, noted for 1 month. He is previously healthy with no traumatic accident to his left wrist. Roentgenogram revealed loss of radial-lunate joint space and mild

40 Wei Cheng Lai et al. / Tungs Medical Journal 11 (2017) 39-43 sclerotic change over lunate bone (Fig 1a).

It is a Lichtman stage II disease, where the outline is normal, but definite density changes are present within the lunate. Operative treatment with volar wrist approach was performed.

2 40 Wei Cheng Lai et al. / Tungs Medical Journal 11 (2017) sclerotic change over lunate bone (Fig 1a). His magnetic resonance imaging (MRI) demonstrated long T2 signal on the lunate with loss of normal short T1 signal (Fig 2a, b), suggesting osteonecrosis of lunate bone (Kienböck s disease). It is a Lichtman stage II disease, where the outline is normal, but definite density changes are present within the lunate. Operative treatment with volar wrist approach was performed. Necrotic bone over lunate bone near the radial-lunate joint was carefully debrided with preservation of general structure of the bone. After wound closure, we applied external fixator device commonly used for operative reduction of distal radial fracture. Some distraction force was applied before tightening down the external fixation device. The patient was discharged the next day. The external fixator device was removed 2 months postoperatively. Due to extended joint fixation, some loss of range of motion and disuse osteoporosis was noted on follow-up roentgenogram. However, improved contour of radial-lunate joint line is observed (Fig 1b). Rehabilitation program followed and on the third month follow-up, the patient revealed pain free wrist with good range of motion and grip strength. Discussion The goals of treating Kienböck s disease include prevention of further osteonecrosis and collapse and restoration of lunate to normal alignment and function [10]. Conservative treatment with wrist immobilization such as casting may be considered for early stage disease (Lichtman stage I or II). However, with unpredictable outcome and extended length of immobilization (may require 4 months or more), it is quite unacceptable for patients [11]. Joint leveling procedures include ulnar lengthening and radial shortening and usually are indicated for Lichtman stage I through IIIA Kienböck s disease, with a negative ulnar variance and without degenerative changes in the radiolunate or capitolunate joints [11]. However, as reported by Ryogo Nakamura [12], not all patients with Kienböck s disease have negative variance. Patients with ulnar-positive or ulnar-neutral variance cannot undergo a joint leveling procedure. Sources of vascularized graft for lunate revascularization procedures include the distal radius based on the pronate quadratus, the pisiform as a pedicle (a) (b) Fig. 1 (a) Initial presentation. Roentgenogram revealed loss of radial-lunate joint space and mild sclerotic change over lunate bone. (b) Two months post-operative, before external fixator removal. Disuse osteoporosis was noted on follow-up roentgenogram. However, improved contour of radial-lunate joint line is observed.

Wei Cheng Lai et al. / Tungs Medical Journal 11 (2017) 39-43 41 graft, and various other grafts from the distal radius, second metacarpal, and pisiform [11].

Moreover, most reports reflect that the promising early radiographic changes may not persist over time, and in many patients there is further deterioration in radiographic and clinical results [11].

3 Wei Cheng Lai et al. / Tungs Medical Journal 11 (2017) graft, and various other grafts from the distal radius, second metacarpal, and pisiform [11]. However, such procedures put a great demand on technical and surgical expertise [13], and cannot be reliably performed by surgeons with less experience. Moreover, most reports reflect that the promising early radiographic changes may not persist over time, and in many patients there is further deterioration in radiographic and clinical results [11]. Temporary internal fixation of the scaphotrapezio-trapezoidal (STT) joint for the treatment of Kienböck s disease is another viable option for early stage diseases [14,15]. This is slightly less effective in reducing the load across the lunate with progressive ulnar deviation of the wrist. This procedure aims to decompress the lunate by shifting the load transferred through the wrist via the capitoscaphoid and radioscaphoid joints. It also may prevent the radiocarpal arthrosis that is secondary to rotary subluxation of the scaphoid following lunate collapse [14]. Complications occurring during temporary fixation included K-wire loosening and breakage. However, without associated vascular procedure, the positive effect of STT is still uncertain. Advanced stage Kienböck s disease leads to destruction of other carpal bones and severe loss of wrist function. In advanced stages, patient may require extensive procedure such as proximal row carpectomy. However, proximal row carpectomy may not be appropriate for Kienböck s disease since lunate collapse damages joint surfaces of the capitate and radius. Wrist arthrodesis is indicated in persons who use their hands for heavy labor, have severe degenerative changes, or fail to improve following other surgical procedures. Our aim is to hold the progression of Kienböck s disease and hopefully stop extensive destruction of the wrist joint. In order to find a less technically demanding procedure for the treatment of Kienböck s disease and prevent associated complications, we applied the principle of distraction histiogenesis as used in Perthes disease in children [8,9]. Debridement of necrotic bone will start the healing process where osteocytes become viable. Distraction of the carpus will unload and relief the injured bone, increase vascularity of the region and help maintain the health of the lunate bone. The combination will provide a biologically viable bed for the surviving osteocytes. In addition, lunate height is maintained while healing occurs. In our particular case, the patient s roentgenogram did not reveal ulnar-negative variance, thus the (a) (b) Fig. 2 (a) MRI of wrist demonstrated loss of normal short T1 signal on the lunate, suggesting osteonecrosis of lunate bone. (b) MRI of wrist demonstrated loss of normal short T1 signal on the lunate, suggesting osteonecrosis of lunate bone.

4 42 Wei Cheng Lai et al. / Tungs Medical Journal 11 (2017) patient cannot undergo a joint leveling procedure. Due to the relatively early stage of the disease (Lichtman stage II), we decided to utilize a simple and familiar procedure that young orthopedic surgeon can comfortably perform instead of technically demanding procedures such as vascularized graft. The debridement procedure uses standard volar wrist approach, which is safe and familiar to general orthopedic surgeons. Distraction was done after wound closure with a simple external fixation device commonly used for treatment of distal radial fracture. The application of such external fixation device is familiar to most orthopedic surgeons. This procedure is not without complications. The extended length of fixing a wrist with an external fixator device produces wrist joint stiffness with limited mobility. Although rehabilitation program should be followed afterwards, sometimes patient cannot regain his previous level of strength or mobility, resulting in loss of wrist function. Another common complication with using external fixator device is pin tract infection. With careful patient education and wound care, such complication still occurs from time to time. Oral antibiotics combined with local wound debridement should allow retaining of the external fixator device. In conclusion, our method of treating Kienböck s disease resulted in good symptomatic relief, with return to normal activities. It is unfortunate that Kienböck s disease is a rare occurrence. Our method of treatment requires further experience and research. To our knowledge, there were only a few reports on the treatment of Kienböck s disease with distraction histiogenesis [10]. Using the distraction principle, osteonecrosis of other bones may also benefit from this method of treatment. References 1. D Agostino C, Romeo P, Amelio E, Sansone V. Effectiveness of ESWT in the treatment of Kienböck s disease. Ultrasound Med Biol. 2011; 37(9): Salmon J, Stanley JK, Trail IA. Kienböck s disease: conservative management versus radial shortening. J Bone Joint Surg Br. 2000; 82(6): Kawoosa AA, Dhar SA, Mir MR, Butt MF. Distraction osteogenesis for ulnar lengthening in Kienbock s disease. Int Orthop. 2007; 31(3): Shin AY, Bishop AT. Pedicled vascularized bone grafts for disorders of the carpus: scaphoid nonunion and Kienbock s disease. J Am Acad Orthop Surg. 2002; 10(3): Graner O, Lopes EI, Carvalho BC, Atlas S. Arthrodesis of the carpal bones in the treatment of Kienböck s disease, painful ununited fractures of the navicular and lunate bones with avascular necrosis, and old fracture-dislocations of carpal bones. J Bone Joint Surg Am. 1966; 48(4): Croog AS, Stern PJ. Proximal row carpectomy for advanced Kienböck s disease: average 10-year follow-up. J Hand Surg Am. 2008; 33(7): Evans G, Burke FD, Barton NJ. A comparison of conservative treatment and silicone replacement arthroplasty in Kienböck s disease. J Hand Surg Br. 1986; 11(1): Segev E, Ezra E, Wientroub S, Yaniv M. Treatment of severe late onset Perthes disease with soft tissue release and articulated hip distraction: early results. J Pediatr Orthop B. 2004; 13(3): Satoshi O, Natsuo Y, Takanobu N, Shinji H, Hideki Y. Articular distraction with external fixator for collapsed femoral head in Perthes disease. J Jpn Assocf Ext Fix Limb Lengthening. 2001; 12: L. 10. Meena D, Saini N, Kundanani V, Chaudhary L, Meena D. Distraction histiogenesis for treatment of Kienbock s disease: A 2- to 8-year follow-up. Indian J Orthop. 2009; 43(2): Philip E, Wright II. Wrist Disorders: Fractures and dislocations of the carpal bones, including Kienböck disease. In: Canale ST, Beaty JH, eds. Campbell s Operative Orthopaedics. 11th ed, vol. 4. Philadelphia: Mosby; p Nakamura R, Tsuge S, Watanabe K, Tsunoda K. Radial wedge osteotomy for Kienböck disease. J Bone Joint Surg Am. 1991; 73(9): Sheetz KK, Bishop AT, Berger RA. The arterial blood supply of the distal radius and ulna and its potential use in vascularized pedicled bone grafts. J Hand Surg Am. 1995; 20(6): Yajima H, Ono H, Tamai S. Temporary internal fixation of the scaphotrapezio-trapezoidal joint for the treatment of Kienböck s disease: a preliminary study. J Hand Surg Am. 1998; 23(3): Watson HK, Monacelli DM, Milford RS, Ashmead D IV. Treatment of Kienböck s disease with scaphotrapeziotrapezoid arthrodesis. J Hand Surg Am. 1996; 21(1): 9-15.

5 Wei Cheng Lai et al. / Tungs Medical Journal 11 (2017) 病例報告 : 以牽引骨增生之方法治療第二期的月狀骨無菌性壞死 * 摘要月狀骨無菌性壞死是手腕的一種罕見的疾病, 最終會造成腕部月狀骨完全壞死如不積極治療, 這種疾病通常會導致月狀骨碎裂, 塌陷與腕骨縮短然而, 現階段關於治療沒有達成共識, 方法包括保守或手術治療各種手術治療的方法已有多種嚐試, 各種手術方法也有達成其不同的預後在此疾病的不同階段手術治療的方法也不盡相同在此篇文章, 我們報告了一位月狀骨無菌性壞死第二期病患的手術治療經驗我們手術進行月狀骨死骨清創, 接著使用外固定支架來達到牽引骨增生的作用我們的結果顯示, 這位病患在手腕的力量和活動性都有不錯的改善, 有利於功能的恢復應行進一步研究來了解這種治療方法的全部潛力關鍵詞 : 月狀骨無菌性壞死牽引成骨術骨壞死 * 通訊作者 :

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