282 Case report
Vascularized bone transfer for the treatment of advancedstage juvenile lunatomalacia: a report of two cases Sebastian Farr, Rudolf Ganger and Werner Girsch We present the first two cases of juvenile patients, with advanced-stage lunatomalacia (Lichtman stage IIIB) in the literature, who were successfully treated with vascularized bone transfers. A corticoperiosteal metacarpal II bone graft was used in both patients, accompanied by an external monorail fixateur that was mounted for 6 weeks to achieve temporary wrist distraction. At 1-year follow-up, the patients had no residual pain, full wrist range of motion, and proper radiologic remodeling of the collapsed lunate. Surgically diminished compression forces at wrist level, temporary gain of joint width, as well as high juvenile potential for remodeling might have enhanced lunate
c 2014 revascularization. J Pediatr Orthop B 23:282–284 Wolters Kluwer Health | Lippincott Williams & Wilkins.
Introduction
(701) on physical examination. Despite her advanced carpal destruction showing lunate fractures with significant height reduction, we aimed to avoid proximal row carpectomy or any other definitive arthrodesis because of her age. We thus performed a vascularized bone transfer using a corticoperiosteal metacarpal II bone graft, which was transferred by means of a dorsal wrist approach [3,4]. After an S-shaped incision, the second dorsal metacarpal artery, which originates from the anatomic snuffbox, was identified. The vessel was followed and dissected to the level of its insertion into the metacarpal base. Thereafter, a 5 3 mm corticoperiosteal bone graft was defined, carefully raised using an osteotome, and graft vitality was confirmed after tourniquet deflation. The whole bone block including the vascular pedicle was thereafter mobilized and reverted under the wrist extensor tendons. The graft was inserted in the lunate after careful curettage, with the wrist being held in flexion to achieve optimal positioning. As the bone graft was inserted in a press-fit-like manner, no osteosynthetic fixation was necessary. An external monorail fixateur (Hoffmann; Stryker Inc., Kalamazoo, Michigan, USA) was then mounted to achieve temporary wrist distraction (5 mm) resulting in decreased compression forces (Fig. 1d). In addition, we aimed for ulnar corrective and radius shortening osteotomies to achieve neutral variance at the wrist level. Both joint levelling procedures were performed in the same session before graft transfer. The fixateur was removed 6 weeks postoperatively. Lunate remodeling was visible throughout the postoperative course, starting at 4 months postoperatively. Further postoperative radiographs revealed good graft integration and complete restoration of the bone structure was observed at 1-year follow-up (Fig. 1e and f). The patient was satisfied, showed full wrist range of motion without any pain, and was fully capable of handling even heavier loads including axial load bearing.
Lunatomalacia or avascular necrosis of the lunate in children and adolescents can eventually cause severe sequelae in the immature wrist joint. Although earlier stages in pediatric patients may be successfully treated using nonoperative treatment, several surgical methods have been described for advanced cases [1]. They comprise radial shortening osteotomies, temporary fixation of the scaphotrapeziotrapezoidal joint, and capitate shortening. Vascularized bone transfers, also primarily published for adult cases, aim to directly restore blood supply and hence restoration of lunate (micro)architecture may be expected. However, if lunatomalacia proceeds, showing rather massive signs of carpal destruction (Lichtman stages IIIB or IV), salvage procedures such as proximal row carpectomy may finally be indicated for adult cases. Despite all different surgical methods, literature on clinical results and treatment algorithms in immature patients (< 15 years) is scarce. Successful results have been demonstrated after conservative treatment in patients under the age of 8 and 12 years [1,2]. However, most of these cases were still relatively stable with regard to carpal height and lunate integrity. Thus, to the best of our knowledge, we did not find any reports about juveniles treated for advanced lunate destruction by vascularized means as performed in this report. We present two juvenile patients, with advancedstage lunatomalacia, who were successfully treated with vascularized bone transfers and temporary wrist distraction.
Case report The first case, a 15-year-old girl, was referred to our tertiary care center showing massive lunate deformation (stage IIIB) and ulna hypoplasia (stage I) on recent radiographs (Fig. 1a–c). Although she was reported to have suffered from pain for almost 1 year, she actually showed stressdependent pain besides minor reduction of wrist extension c 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins 1060-152X
Journal of Pediatric Orthopaedics B 2014, 23:282–284 Keywords: external monorail fixateur, juvenile lunatomalacia, metacarpal bone graft, temporary wrist distraction, vascularized bone transfer Department of Pediatric Orthopaedics, Deformity Correction and Adult Foot and Ankle Surgery, Orthopaedic Hospital Speising, Vienna, Austria Correspondence to Sebastian Farr, MD, Department of Pediatric Orthopaedics, Deformity Correction and Adult Foot and Ankle Surgery, Orthopaedic Hospital Speising, Speisinger Strasse 109, 1130 Vienna, Austria Tel: + 43 1 80182 1610; fax: + 43 1 80182 1644; e-mail: [email protected]
DOI: 10.1097/BPB.0000000000000013
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Advanced-stage juvenile lunatomalacia Farr et al. 283
Fig. 1
Case 1: (a) Preoperative radiograph showing ulnar hypoplasia, massive lunate deformation, and reduction of carpal height. (b) MRI sequences confirmed the avascular necrosis of the bone. (c) Computed tomography reveals partial fragmentation of the lunate in the lateral view. (d) Intraoperative situation after mounting of external fixateur and ulnar corrective and radius shortening osteotomy to achieve neutral variance at the wrist level. (e, f) Radiography and computed tomography demonstrating lunate healing and consolidation of carpal height in the later postoperative course.
Fig. 2
Case 2: (a) Lunate destruction is visible at the triquetral border. (b, c) MRI and computed tomography confirmed the bone necrosis revealing a shelllike, fractured lunate at risk for carpal collapse. (d–f) After short-term wrist distraction, 1-year follow-up demonstrated complete bone remodeling.
The second patient, a 15-year-old boy, showed a relatively similar clinical course. He presented with marked reduction of wrist motion (601 flexion and extension) showing a maximum of pain during wrist extension since 6 months. His recent radiographs revealed lunatomalacia stage IIIB, which was accordingly addressed with a vascularized corticoperiosteal metacarpal II bone graft, accompanied by external monorail fixateur as mentioned before (Fig. 2a–c). No accompanying procedures were performed because of the regular anatomy at the wrist joint level. After the fixateur was removed, he was treated in the same manner as case 1 with occupational therapy, load restriction, and activity modification. Radiographs more than 1 year after the procedure showed full restoration of lunate structure (Fig. 2d–f). Clinical evalua-
tion revealed improved motion without any tenderness or provocative pain. Moreover, the patient was satisfied and resumed all his previous activities without restrictions.
Discussion Lunatomalacia in its heaviest form may lead to pancarpal arthritis, which in turn is a relevant concern, especially if the patient is of young age. To the best of our knowledge we did not find any treatment algorithms for advancedstage juvenile lunatomalacia because of paucity in the current literature. The only study found, including a vascularized bone transfer in a child (pisiform transfer), reported a successful MRI outcome but significant wrist pain 8.5 years postoperatively [5]. We do not agree with
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
284
Journal of Pediatric Orthopaedics B 2014, Vol 23 No 3
other authors who stated that pediatric lunatomalacia is a self-limiting, nonprogressive condition as carpal integrity in our cases was heavily disturbed, showing massive destruction and even fragmentation in both instances [2]. As a consequence, we considered reconstruction of the avascular lunate by vascularized means to prevent the bone and eventually the carpus from further collapse. It remains of course unclear, whether temporary wrist distraction significantly contributed to the healing process. However, we believe that surgical decrease of compression forces in case 1 (joint levelling surgery), temporary gain of joint width by the fixator, as well as the juvenile potential for remodeling might have enhanced lunate revascularization. As highlighted by Dias and Lunn [6] in a recent review, there is much uncertainty in this disease concerning not only natural history but also optimal treatment and surgery to prevent the lunate from collapsing. Particularly in children and adolescents, where biology might be better capable of dealing with these issues, current treatment is still experimental and often ‘custom-made’. One should, of course, address the present anatomic differences (i.e. ulnar negative variance) by established means. Nevertheless, we believe that the disastrous situation in our patients necessitated a combination of procedures to achieve a good result in
this short-term period. Whether the vascularized graft, the graft and distraction, or even distraction alone was the main factor for healing remains unknown. Longer follow-up of our cases is necessary to confirm remodeling in the long term as are large, homogeneous studies of such pediatric cohorts.
Acknowledgements Conflicts of interest
There are no conflicts of interest.
References 1 2 3
4
5
6
Irisarri C, Kalb K, Ribak S. Infantile and juvenile lunatomalacia. J Hand Surg Eur Vol 2010; 35:544–548. Cvitanich M, Solomons M. Juvenile lunatomalacia is this Kienbo¨ck’s disease? J Hand Surg Br 2004; 29:288–292. Sawaizumi T, Nanno M, Nanbu A, Ito H. Vascularised bone graft from the base of the second metacarpal for refractory nonunion of the scaphoid. J Bone Joint Surg Br 2004; 86:1007–1012. Makino M. Vascularized metacarpal bone graft for scaphoid non-union and Kienbo¨ck’s disease. J Reconstr Microsurg 2000; 16:261–266, discussion 266–268. Kalb K, Pillukat T, Schmitt R, Prommersberger KJ. Kienbo¨ck’s disease in paediatric and juvenile patients. Handchir Mikrochir Plast Chir 2010; 42:187–197. Dias JJ, Lunn P. Ten questions on Kienbo¨ck’s disease of the lunate. J Hand Surg Eur Vol 2010; 35:538–543.
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Vascularized bone transfer for the treatment of advancedstage juvenile lunatomalacia: a report of two cases Sebastian Farr, Rudolf Ganger and Werner Girsch We present the first two cases of juvenile patients, with advanced-stage lunatomalacia (Lichtman stage IIIB) in the literature, who were successfully treated with vascularized bone transfers. A corticoperiosteal metacarpal II bone graft was used in both patients, accompanied by an external monorail fixateur that was mounted for 6 weeks to achieve temporary wrist distraction. At 1-year follow-up, the patients had no residual pain, full wrist range of motion, and proper radiologic remodeling of the collapsed lunate. Surgically diminished compression forces at wrist level, temporary gain of joint width, as well as high juvenile potential for remodeling might have enhanced lunate
c 2014 revascularization. J Pediatr Orthop B 23:282–284 Wolters Kluwer Health | Lippincott Williams & Wilkins.
Introduction
(701) on physical examination. Despite her advanced carpal destruction showing lunate fractures with significant height reduction, we aimed to avoid proximal row carpectomy or any other definitive arthrodesis because of her age. We thus performed a vascularized bone transfer using a corticoperiosteal metacarpal II bone graft, which was transferred by means of a dorsal wrist approach [3,4]. After an S-shaped incision, the second dorsal metacarpal artery, which originates from the anatomic snuffbox, was identified. The vessel was followed and dissected to the level of its insertion into the metacarpal base. Thereafter, a 5 3 mm corticoperiosteal bone graft was defined, carefully raised using an osteotome, and graft vitality was confirmed after tourniquet deflation. The whole bone block including the vascular pedicle was thereafter mobilized and reverted under the wrist extensor tendons. The graft was inserted in the lunate after careful curettage, with the wrist being held in flexion to achieve optimal positioning. As the bone graft was inserted in a press-fit-like manner, no osteosynthetic fixation was necessary. An external monorail fixateur (Hoffmann; Stryker Inc., Kalamazoo, Michigan, USA) was then mounted to achieve temporary wrist distraction (5 mm) resulting in decreased compression forces (Fig. 1d). In addition, we aimed for ulnar corrective and radius shortening osteotomies to achieve neutral variance at the wrist level. Both joint levelling procedures were performed in the same session before graft transfer. The fixateur was removed 6 weeks postoperatively. Lunate remodeling was visible throughout the postoperative course, starting at 4 months postoperatively. Further postoperative radiographs revealed good graft integration and complete restoration of the bone structure was observed at 1-year follow-up (Fig. 1e and f). The patient was satisfied, showed full wrist range of motion without any pain, and was fully capable of handling even heavier loads including axial load bearing.
Lunatomalacia or avascular necrosis of the lunate in children and adolescents can eventually cause severe sequelae in the immature wrist joint. Although earlier stages in pediatric patients may be successfully treated using nonoperative treatment, several surgical methods have been described for advanced cases [1]. They comprise radial shortening osteotomies, temporary fixation of the scaphotrapeziotrapezoidal joint, and capitate shortening. Vascularized bone transfers, also primarily published for adult cases, aim to directly restore blood supply and hence restoration of lunate (micro)architecture may be expected. However, if lunatomalacia proceeds, showing rather massive signs of carpal destruction (Lichtman stages IIIB or IV), salvage procedures such as proximal row carpectomy may finally be indicated for adult cases. Despite all different surgical methods, literature on clinical results and treatment algorithms in immature patients (< 15 years) is scarce. Successful results have been demonstrated after conservative treatment in patients under the age of 8 and 12 years [1,2]. However, most of these cases were still relatively stable with regard to carpal height and lunate integrity. Thus, to the best of our knowledge, we did not find any reports about juveniles treated for advanced lunate destruction by vascularized means as performed in this report. We present two juvenile patients, with advancedstage lunatomalacia, who were successfully treated with vascularized bone transfers and temporary wrist distraction.
Case report The first case, a 15-year-old girl, was referred to our tertiary care center showing massive lunate deformation (stage IIIB) and ulna hypoplasia (stage I) on recent radiographs (Fig. 1a–c). Although she was reported to have suffered from pain for almost 1 year, she actually showed stressdependent pain besides minor reduction of wrist extension c 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins 1060-152X
Journal of Pediatric Orthopaedics B 2014, 23:282–284 Keywords: external monorail fixateur, juvenile lunatomalacia, metacarpal bone graft, temporary wrist distraction, vascularized bone transfer Department of Pediatric Orthopaedics, Deformity Correction and Adult Foot and Ankle Surgery, Orthopaedic Hospital Speising, Vienna, Austria Correspondence to Sebastian Farr, MD, Department of Pediatric Orthopaedics, Deformity Correction and Adult Foot and Ankle Surgery, Orthopaedic Hospital Speising, Speisinger Strasse 109, 1130 Vienna, Austria Tel: + 43 1 80182 1610; fax: + 43 1 80182 1644; e-mail: [email protected]
DOI: 10.1097/BPB.0000000000000013
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Advanced-stage juvenile lunatomalacia Farr et al. 283
Fig. 1
Case 1: (a) Preoperative radiograph showing ulnar hypoplasia, massive lunate deformation, and reduction of carpal height. (b) MRI sequences confirmed the avascular necrosis of the bone. (c) Computed tomography reveals partial fragmentation of the lunate in the lateral view. (d) Intraoperative situation after mounting of external fixateur and ulnar corrective and radius shortening osteotomy to achieve neutral variance at the wrist level. (e, f) Radiography and computed tomography demonstrating lunate healing and consolidation of carpal height in the later postoperative course.
Fig. 2
Case 2: (a) Lunate destruction is visible at the triquetral border. (b, c) MRI and computed tomography confirmed the bone necrosis revealing a shelllike, fractured lunate at risk for carpal collapse. (d–f) After short-term wrist distraction, 1-year follow-up demonstrated complete bone remodeling.
The second patient, a 15-year-old boy, showed a relatively similar clinical course. He presented with marked reduction of wrist motion (601 flexion and extension) showing a maximum of pain during wrist extension since 6 months. His recent radiographs revealed lunatomalacia stage IIIB, which was accordingly addressed with a vascularized corticoperiosteal metacarpal II bone graft, accompanied by external monorail fixateur as mentioned before (Fig. 2a–c). No accompanying procedures were performed because of the regular anatomy at the wrist joint level. After the fixateur was removed, he was treated in the same manner as case 1 with occupational therapy, load restriction, and activity modification. Radiographs more than 1 year after the procedure showed full restoration of lunate structure (Fig. 2d–f). Clinical evalua-
tion revealed improved motion without any tenderness or provocative pain. Moreover, the patient was satisfied and resumed all his previous activities without restrictions.
Discussion Lunatomalacia in its heaviest form may lead to pancarpal arthritis, which in turn is a relevant concern, especially if the patient is of young age. To the best of our knowledge we did not find any treatment algorithms for advancedstage juvenile lunatomalacia because of paucity in the current literature. The only study found, including a vascularized bone transfer in a child (pisiform transfer), reported a successful MRI outcome but significant wrist pain 8.5 years postoperatively [5]. We do not agree with
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
284
Journal of Pediatric Orthopaedics B 2014, Vol 23 No 3
other authors who stated that pediatric lunatomalacia is a self-limiting, nonprogressive condition as carpal integrity in our cases was heavily disturbed, showing massive destruction and even fragmentation in both instances [2]. As a consequence, we considered reconstruction of the avascular lunate by vascularized means to prevent the bone and eventually the carpus from further collapse. It remains of course unclear, whether temporary wrist distraction significantly contributed to the healing process. However, we believe that surgical decrease of compression forces in case 1 (joint levelling surgery), temporary gain of joint width by the fixator, as well as the juvenile potential for remodeling might have enhanced lunate revascularization. As highlighted by Dias and Lunn [6] in a recent review, there is much uncertainty in this disease concerning not only natural history but also optimal treatment and surgery to prevent the lunate from collapsing. Particularly in children and adolescents, where biology might be better capable of dealing with these issues, current treatment is still experimental and often ‘custom-made’. One should, of course, address the present anatomic differences (i.e. ulnar negative variance) by established means. Nevertheless, we believe that the disastrous situation in our patients necessitated a combination of procedures to achieve a good result in
this short-term period. Whether the vascularized graft, the graft and distraction, or even distraction alone was the main factor for healing remains unknown. Longer follow-up of our cases is necessary to confirm remodeling in the long term as are large, homogeneous studies of such pediatric cohorts.
Acknowledgements Conflicts of interest
There are no conflicts of interest.
References 1 2 3
4
5
6
Irisarri C, Kalb K, Ribak S. Infantile and juvenile lunatomalacia. J Hand Surg Eur Vol 2010; 35:544–548. Cvitanich M, Solomons M. Juvenile lunatomalacia is this Kienbo¨ck’s disease? J Hand Surg Br 2004; 29:288–292. Sawaizumi T, Nanno M, Nanbu A, Ito H. Vascularised bone graft from the base of the second metacarpal for refractory nonunion of the scaphoid. J Bone Joint Surg Br 2004; 86:1007–1012. Makino M. Vascularized metacarpal bone graft for scaphoid non-union and Kienbo¨ck’s disease. J Reconstr Microsurg 2000; 16:261–266, discussion 266–268. Kalb K, Pillukat T, Schmitt R, Prommersberger KJ. Kienbo¨ck’s disease in paediatric and juvenile patients. Handchir Mikrochir Plast Chir 2010; 42:187–197. Dias JJ, Lunn P. Ten questions on Kienbo¨ck’s disease of the lunate. J Hand Surg Eur Vol 2010; 35:538–543.
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
