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Radical Resection of a Massive Venous Malformation of the Thumb and Immediate Reconstruction With a Microsurgical Toe Transfer
Radical Resection of a Massive Venous Malformation of the Thumb and Immediate Reconstruction With a Microsurgical Toe Transfer Brian Carlsen, MD, Neil F. Jones, MD From the UCLA Hand Center, Los Angeles, CA.
A massive venous malformation involving the entire thumb, first web space, and index finger in a young child was treated by radical resection of the venous malformation, including amputation of the thumb distal to the mid-metacarpal and immediate reconstruction with a microsurgical second toe transfer. Five years after surgery, the patient has excellent function of the hand and no evidence of recurrence of the malformation. This case is unique in two respects: first, the venous malformation was of massive size, and second, the unusual venous malformation required total amputation of the thumb and immediate reconstruction with a complete toe transfer. (J Hand Surg 2007;32A:1587–1591. Copyright © 2007 by the American Society for Surgery of the Hand.) Key words: Venous malformations, vascular malformations, toe transplantation, congenital hand deformities, microsurgery.
ascular malformations include capillary, lymphatic, venous, and arteriovenous malformations (AVM). The most common type in the upper extremity is the venous malformation (VM), which makes 46% of all malformations.1 Vascular malformations are notoriously difficult to treat. Initial treatment is conservative, including compression therapy and injection sclerotherapy. Indications for surgical intervention include pain, bleeding, nerve compression, infection, and functional impairment. Most VMs will require surgical resection for one of these indications. Recurrence is common, however, especially in diffuse lesions that involve 2 or more tissue types as opposed to local lesions that involve only a single tissue type, such as muscle.2,3 Recurrence is probably inevitable if any portion of the malformation is left behind after surgical resection,4,5 and therefore a radical surgical approach has been advocated.1,2,6 Hill et al2 reported 15 vascular malformations of the upper extremity treated by radical resection and still had a 47% recurrence rate. Yoshii reported recurrence of 2 of 6 AVMs treated with ligation and excision, but no recurrence in 4 patients treated with radical excision followed by flap reconstruction.5 Other case reports and small series
V
have suggested that radical excision and free flap reconstruction may prevent recurrences.7–12 Radical resection of vascular malformations may result, however, in an extensive anatomic and functional defect. We report a young child with a massive VM involving the thumb, first web space, and index finger treated by radical resection and immediate reconstruction with a second toe transfer.
Case Report This young boy was born with a congenital vascular malformation involving the left thumb, first web space, and index finger, which rapidly enlarged during his first year of life to envelop the entire left thumb, first web space, and radial aspect of the index finger (Fig. 1). It completely impaired the function of his left hand. He could not pinch or grip because of the bulk of the mass and could not lift his arm because of the weight of the hand. There was no palpable thrill or appreciable bruit. A magnetic resonance imaging (MRI) scan performed at 13 days of age at an outside institution suggested an AVM arising from the deep palmar arch. Subsequent MRI scan and angiography showed vascular lakes that were more consistent with a VM (Fig. 2). Initial treatment The Journal of Hand Surgery
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The Journal of Hand Surgery / Vol. 32A No. 10 December 2007
Figure 2. Angiogram showing pooling of contrast in vascular lakes.
radial digital artery to the index finger was involved and resected; however, the radial digital nerve was spared (Fig. 3A). He had immediate reconstruction with a microsurgical second toe transfer from his right foot. Skeletal fixation was achieved by inserting the metatarsal bone into the medullary canal of the thumb metacarpal and secured with 26-gauge intraosseous wiring (Fig. 4). Standard microsurgical techniques were used to coapt the digital nerves and anastomose one dorsal vein and a vein graft from the radial artery in the anatomic snuffbox to the first plantar metatarsal artery. The flexor digitorum longus tendon to the Figure 1. Palmar (A) and dorsal (B) views of the massive VM involving the left thumb, first web space, and index finger.
was nonsurgical, including intravenous steroid infusion and 4 attempts at direct injection sclerotherapy. The second injection was complicated by necrosis of the thenar skin and required a 10-day hospitalization for wound care and observation. The sclerosing agents seemed to diminish the vascular nature of the lesion but did not reduce its size or weight. Because the malformation totally impaired the function of his hand and because of the failure of injection sclerotherapy, he had radical resection of the VM at 42 months of age. Definitive resection required amputation of the thumb at the metacarpophalangeal joint and resection of the malformation right up to the level of the dermis on the skin flaps in the first web space and index finger (Fig. 3). The
Figure 3. Palmar (A) and dorsal (B) views of the hand after radical resection, just before immediate reconstruction with a microsurgical second toe transfer.
Carlsen and Jones / Toe Transfer for Venous Malformation
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Vascular malformations are classified as high-flow (arterial) or low-flow (venous or capillary) malformations. Nearly 90% of vascular malformations are low-flow lesions and half of these are VMs.1 They are further divided into local and diffuse malformations. Local lesions involve only one tissue type, whereas diffuse lesions involve two or more tissue types.3 In this case, the lesion was initially diagnosed as an AVM on MRI at 13 days of life; the MRI was performed and read at an outside institution. Subsequent studies, including angiography, were more consistent with a VM. The diagnosis of vascular lesions can be quite difficult; however, MRI with dynamic contrast-enhancement remains the best technique for definitive diagnosis.14
Figure 4. Radiograph 2 years after surgery demonstrating skeletal fixation of the transferred toe metatarsal to the thumb metacarpal with 90 –90 intraosseous wiring.
toe was repaired to the flexor pollicis longus tendon, and the extensor digitorum longus tendon was sutured to the extensor pollicis longus tendon. Histological examination of the lesion was consistent with a diffuse VM. The patient’s postoperative course was uncomplicated, and he was discharged home 9 days after surgery. Follow-up evaluation 5 years after surgery shows excellent healing of the toe transfer, and the patient uses the thumb normally for grip and pinch functions (Fig. 5). He is able to oppose to all four fingertips and has good abduction, but he cannot fully pronate. Radiographs demonstrate solid osteosynthesis to the thumb metacarpal and similar growth compared to the second toe on the contralateral left foot (Fig. 4). There is no recurrence of the VM.
Discussion Vascular anomalies, hemangiomas, and vascular malformations together make up the fourth most common types of mass of the hand.2,4 Nomenclature of vascular lesions remains problematic and has resulted in confusion and misdiagnosis. Vascular malformations are differentiated from hemangiomas based on cellular characteristics and have different biological behavior.13 Hemangiomas are common in infancy and exhibit a period of rapid growth followed by slow involution around 5 to 7 years. Vascular malformations are not neoplasms but are biologically quiescent lesions that share the same growth rate as normal tissues.
Figure 5. (A, B) The reconstructed hand at 4 years after surgery, showing excellent function and no evidence of recurrence.
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The Journal of Hand Surgery / Vol. 32A No. 10 December 2007
Malformations can become symptomatic with pain, heaviness, thrombosis, skin maceration, infection, distal ischemia, and functional impairment. Initial management is nonsurgical and includes application of compression garments, laser therapy, and injection sclerotherapy. Ligation of feeder vessels should be avoided, as this is unlikely to effectively treat the lesion and may cause distal ischemia and necrosis. Surgical resection is associated with significant morbidity and a significant risk of recurrence.1,2 Hill et al reported a 47% recurrence rate even after radical resection; however, they felt that excision was incomplete in 6 of the 7 recurrences.2 Despite the risks, surgical resection is often required for definitive treatment and results in symptomatic improvement in most patients. In this case, injection sclerotherapy was performed 4 times, but this treatment did not effectively treat the malformation and was associated with skin necrosis. After 4 treatments, it was obvious that the lesion was not going to respond to nonsurgical treatments and radical resection would be necessary. The treatment of malformations, whether surgical or nonsurgical, should be approached cautiously and treatment planned in consultation with interventional radiology colleagues. Upton et al in a review of 270 patients with vascular malformations, advocated a systematic, staged approach to resection from distal to proximal and dorsal to volar, awaiting return of function and softening of tissues prior to reoperation.1 They recommended complete excision of a single anatomic area at each stage. Repeat operations and postoperative immobilization can lead to scarring and stiffness. Given the importance of the thumb in hand function and our confidence in radical resection, immediate reconstruction was felt to be appropriate in this case. Yoshii et al5 first reported that radical resection of vascular malformations of the forearm and hand followed by flap coverage seemed to prevent recurrence, and this has been subsequently confirmed by others.9,11 Koshima et al7 resected a recurrent AVM of the index finger that was reconstructed with an extended wraparound flap, consisting of a wraparound flap combined with a dorsalis pedis flap. Adani et al8 reported resection of a low-flow VM of the distal phalanx of the thumb and reconstruction with a free toe pulp flap. Hattori et al10 also used a wraparound flap combined with a dorsalis pedis flap to cover the tendon and bony skeleton of the thumb after resection of an extensive AVM involving the thumb, index finger, and first web space. Toh et al11
reported 3 patients with AVMs involving the palmar aspect of the index finger, distal pulp of the thumb, and thenar eminence treated by radical resection and free flap coverage using 2 dorsalis pedis flaps and 1 peroneal flap. One patient had an ulcerating distal phalanx of the thumb that was reconstructed with a wraparound flap. Lu et al12 described resection of an AVM involving the thumb, thenar eminence, and first web space, and coverage of the tendon and bony skeleton of the thumb with a wraparound flap and the first web space covered with the extended dorsalis pedis flap. Of these 4 reported cases of VMs involving the thumb, the resultant defects after radical resection were relatively small in 2 patients, involving only the distal phalanx of the thumb and requiring reconstruction with a free toe pulp flap8 and a wraparound flap.11 Even the 2 patients with more extensive defects involving the thumb still had an intact bony skeleton and flexor and extensor tendons and required only soft tissue reconstruction with an extended wraparound flap.10,12 None of these 4 malformations approached the size of the VM presented here, which was truly massive and not only prevented pinch and grip function of the hand but also compromised elbow and shoulder function because of its weight. Additionally, this is a unique case of VM requiring complete amputation of the thumb and immediate reconstruction with an entire toe-to-thumb transfer. Toe-to-hand transfer has become a widely accepted technique for reconstruction of traumatic amputation of the digits since first described by Cobbett and Buncke.15–17 More recently, the indications for toe-to-hand transfers in children are evolving,18 –27 and toe transfers may become more preferable to other alternative techniques such as index finger pollicization, nonvascularized toe phalangeal bone grafting, and distraction lengthening.22 Timing of toe-to-hand transfers in children with congenital hand deformities is an important consideration. Most advocate transfer before age 4,18,23,24 with some recommending surgery around age 2,22,26,27 when the structures are large enough to perform the surgery without too much difficulty. In our case, the futility of nonsurgical management of the malformation became apparent during this optimal time period for toe-to-hand transfer. This case represents successful radical resection of a massive VM requiring amputation of the thumb and immediate reconstruction with a microsurgical second toe transfer. The child has excellent function and no evidence of recurrence after 5 years (Fig. 5).
Carlsen and Jones / Toe Transfer for Venous Malformation Received for publication March 27, 2007; accepted in revised form June 5, 2007. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Corresponding author: Neil F. Jones, MD, UCLA Hand Center, 10945 LeConte Ave., Suite 3355, Los Angeles, CA 90095; e-mail: njones@ mednet.ucla.edu. Copyright © 2007 by the American Society for Surgery of the Hand 0363-5023/07/32A10-0014$32.00/0 doi:10.1016/j.jhsa.2007.06.020
References 1. Upton J, Coombs CJ, Mulliken JB, Burrows PE, Pap S. Vascular malformations of the upper limb: A review of 270 patients. J Hand Surg 1999;24A:1019 –1035. 2. Hill RA, Pho RWH, Kumar VP. Resection of vascular malformations. J Hand Surg 1993;18B:17–21. 3. Szilagy DE, Smith RF, Elliott JP, Hageman JH, Congenital arteriovenous anomalies of the limbs. Arch Surg 1976;111: 423– 429. 4. Bogumill GP, Sullivan DJ, Baker GI. Tumors of the hand. Clin Orthop 1975;108:214 –222. 5. Yoshii T, Tamai S, Fukui N. Microsurgical approach for the hemangiomas and congenital arteriovenous fistulae in forearm and hand. J Jpn Soc Surg Hand 1985;2:736 –741. 6. Upton J, Mulliken JB, Murray JE. Classification and rationale for management of vascular anomalies in the upper extremity. J Hand Surg 1985;10A:970 –975. 7. Koshima I, Soeda S, Murashita T. Extended wrap-around flap for reconstruction of the finger with recurrent arteriovenous malformation. Plas Recon Surg 1993;91:1140 –1144. 8. Adani R, Busa R, Caroli A. Microsurgical approach for a venous malformation of the thumb. J Hand Surg 1997;22B: 30 –31. 9. Yamamoto Y, Ohura T, Minakawa H, Sugihara T, Yoshida T, Nohira K, Shintomi Y. Experience with arteriovenous malformations treated with flap coverage. Plas Recon Surg 1994;94(3):476 – 482. 10. Hattori Y, Doi K, Kawakami F, Watanabe M. Extended wrap-around flap for thumb reconstruction following radical excision of a congenital arteriovenous fistula. J Hand Surg 1998;23B:72–75. 11. Toh S, Tsubo K, Arai H, Harata S. Vascularized free flaps for reconstruction after resection of congenital arteriovenous malformations of the hand. J Recon Microsurg 2000;16(7); 511–517.
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12. Lu LI, Chen DJ, Chen HC, Coessens B. Arteriovenous malformation involving the thumb and hand: radical excision and reconstruction of multiple components. Ann Plast Surg 2002;49:414 – 418. 13. Mulliken JB Glowacki J. Hemangiomas and vascular malformations in infants and children: a classification based on endothelial characteristics. Plast Reconstr Surg 1982;69(3): 412– 422. 14. Vilanova JC, Barcelo J, Villalon M. MR and MR angiography characterization of soft tissue vascular malformations. Curr Probl Diagn Radiol 2004;33(4):161–170. 15. Cobbett JR. Free digital transfer: Report of a case of transfer of a great toe to replace an amputated thumb. J Bone Joint Surg 1969;51B:677– 679. 16. Buncke HJ, McLean DH, George PT, Creech BJ, Chater NL, Commons GW. Thumb replacement: Great toe transplantation by microvascular anastomosis. Br J Plast Surg 1973;26: 194 –201. 17. Morrison WA, O’Brien BM, MacLeod AM. Thumb reconstruction with a free neurovascular wrap-around flap from the big toe. J Hand Surg 1980;5:575–583. 18. Gilbert A. Toe transfers for congenital hand defects. J Hand Surg 1982;7:118 –124. 19. Gilbert A. Reconstruction of congenital hand defects with microvascular toe transfers. Hand Clin 1985;1:351–360. 20. Lister G. Microsurgical transfer of the second toe for congenital deficiency of the thumb. Plas Recon Surg 1988;82: 658 – 665. 21. Buck-Gramcko D. Progress in the treatment of congenital malformations of the hand. World J Surg 1990;14:715–724. 22. Vilkki SK. Advances in microsurgical reconstruction of the congenitally adactylous hand. Clin Orthop 1995;314:45–58. 23. Kay SP Wiberg M. Toe to hand transfer in children, part 1: technical aspects. J Hand Surg 1996;21B:723–734. 24. Kay SP, Wiberg M, Bellew M, Webb F. Toe to hand transfer in children, part 2: functional and psychological aspects. J Hand Surg 1996;21B:735–745. 25. Kay S McGuiness C. Microsurgical reconstruction in abnormalities of children’s hands. Hand Clin 1999;15:563–583. 26. Jones NF, Hansen SL, Bates SJ. Toe-to-hand transfers for congenital anomalies of the hand. Hand Clin 2007;23(1): 129. 27. Chang J Jones NF. Radiographic analysis of growth in pediatric microsurgical toe-to-hand transfers. Plas Recon Surg 2002;109:576 –582.
A massive venous malformation involving the entire thumb, first web space, and index finger in a young child was treated by radical resection of the venous malformation, including amputation of the thumb distal to the mid-metacarpal and immediate reconstruction with a microsurgical second toe transfer. Five years after surgery, the patient has excellent function of the hand and no evidence of recurrence of the malformation. This case is unique in two respects: first, the venous malformation was of massive size, and second, the unusual venous malformation required total amputation of the thumb and immediate reconstruction with a complete toe transfer. (J Hand Surg 2007;32A:1587–1591. Copyright © 2007 by the American Society for Surgery of the Hand.) Key words: Venous malformations, vascular malformations, toe transplantation, congenital hand deformities, microsurgery.
ascular malformations include capillary, lymphatic, venous, and arteriovenous malformations (AVM). The most common type in the upper extremity is the venous malformation (VM), which makes 46% of all malformations.1 Vascular malformations are notoriously difficult to treat. Initial treatment is conservative, including compression therapy and injection sclerotherapy. Indications for surgical intervention include pain, bleeding, nerve compression, infection, and functional impairment. Most VMs will require surgical resection for one of these indications. Recurrence is common, however, especially in diffuse lesions that involve 2 or more tissue types as opposed to local lesions that involve only a single tissue type, such as muscle.2,3 Recurrence is probably inevitable if any portion of the malformation is left behind after surgical resection,4,5 and therefore a radical surgical approach has been advocated.1,2,6 Hill et al2 reported 15 vascular malformations of the upper extremity treated by radical resection and still had a 47% recurrence rate. Yoshii reported recurrence of 2 of 6 AVMs treated with ligation and excision, but no recurrence in 4 patients treated with radical excision followed by flap reconstruction.5 Other case reports and small series
V
have suggested that radical excision and free flap reconstruction may prevent recurrences.7–12 Radical resection of vascular malformations may result, however, in an extensive anatomic and functional defect. We report a young child with a massive VM involving the thumb, first web space, and index finger treated by radical resection and immediate reconstruction with a second toe transfer.
Case Report This young boy was born with a congenital vascular malformation involving the left thumb, first web space, and index finger, which rapidly enlarged during his first year of life to envelop the entire left thumb, first web space, and radial aspect of the index finger (Fig. 1). It completely impaired the function of his left hand. He could not pinch or grip because of the bulk of the mass and could not lift his arm because of the weight of the hand. There was no palpable thrill or appreciable bruit. A magnetic resonance imaging (MRI) scan performed at 13 days of age at an outside institution suggested an AVM arising from the deep palmar arch. Subsequent MRI scan and angiography showed vascular lakes that were more consistent with a VM (Fig. 2). Initial treatment The Journal of Hand Surgery
1587
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The Journal of Hand Surgery / Vol. 32A No. 10 December 2007
Figure 2. Angiogram showing pooling of contrast in vascular lakes.
radial digital artery to the index finger was involved and resected; however, the radial digital nerve was spared (Fig. 3A). He had immediate reconstruction with a microsurgical second toe transfer from his right foot. Skeletal fixation was achieved by inserting the metatarsal bone into the medullary canal of the thumb metacarpal and secured with 26-gauge intraosseous wiring (Fig. 4). Standard microsurgical techniques were used to coapt the digital nerves and anastomose one dorsal vein and a vein graft from the radial artery in the anatomic snuffbox to the first plantar metatarsal artery. The flexor digitorum longus tendon to the Figure 1. Palmar (A) and dorsal (B) views of the massive VM involving the left thumb, first web space, and index finger.
was nonsurgical, including intravenous steroid infusion and 4 attempts at direct injection sclerotherapy. The second injection was complicated by necrosis of the thenar skin and required a 10-day hospitalization for wound care and observation. The sclerosing agents seemed to diminish the vascular nature of the lesion but did not reduce its size or weight. Because the malformation totally impaired the function of his hand and because of the failure of injection sclerotherapy, he had radical resection of the VM at 42 months of age. Definitive resection required amputation of the thumb at the metacarpophalangeal joint and resection of the malformation right up to the level of the dermis on the skin flaps in the first web space and index finger (Fig. 3). The
Figure 3. Palmar (A) and dorsal (B) views of the hand after radical resection, just before immediate reconstruction with a microsurgical second toe transfer.
Carlsen and Jones / Toe Transfer for Venous Malformation
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Vascular malformations are classified as high-flow (arterial) or low-flow (venous or capillary) malformations. Nearly 90% of vascular malformations are low-flow lesions and half of these are VMs.1 They are further divided into local and diffuse malformations. Local lesions involve only one tissue type, whereas diffuse lesions involve two or more tissue types.3 In this case, the lesion was initially diagnosed as an AVM on MRI at 13 days of life; the MRI was performed and read at an outside institution. Subsequent studies, including angiography, were more consistent with a VM. The diagnosis of vascular lesions can be quite difficult; however, MRI with dynamic contrast-enhancement remains the best technique for definitive diagnosis.14
Figure 4. Radiograph 2 years after surgery demonstrating skeletal fixation of the transferred toe metatarsal to the thumb metacarpal with 90 –90 intraosseous wiring.
toe was repaired to the flexor pollicis longus tendon, and the extensor digitorum longus tendon was sutured to the extensor pollicis longus tendon. Histological examination of the lesion was consistent with a diffuse VM. The patient’s postoperative course was uncomplicated, and he was discharged home 9 days after surgery. Follow-up evaluation 5 years after surgery shows excellent healing of the toe transfer, and the patient uses the thumb normally for grip and pinch functions (Fig. 5). He is able to oppose to all four fingertips and has good abduction, but he cannot fully pronate. Radiographs demonstrate solid osteosynthesis to the thumb metacarpal and similar growth compared to the second toe on the contralateral left foot (Fig. 4). There is no recurrence of the VM.
Discussion Vascular anomalies, hemangiomas, and vascular malformations together make up the fourth most common types of mass of the hand.2,4 Nomenclature of vascular lesions remains problematic and has resulted in confusion and misdiagnosis. Vascular malformations are differentiated from hemangiomas based on cellular characteristics and have different biological behavior.13 Hemangiomas are common in infancy and exhibit a period of rapid growth followed by slow involution around 5 to 7 years. Vascular malformations are not neoplasms but are biologically quiescent lesions that share the same growth rate as normal tissues.
Figure 5. (A, B) The reconstructed hand at 4 years after surgery, showing excellent function and no evidence of recurrence.
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The Journal of Hand Surgery / Vol. 32A No. 10 December 2007
Malformations can become symptomatic with pain, heaviness, thrombosis, skin maceration, infection, distal ischemia, and functional impairment. Initial management is nonsurgical and includes application of compression garments, laser therapy, and injection sclerotherapy. Ligation of feeder vessels should be avoided, as this is unlikely to effectively treat the lesion and may cause distal ischemia and necrosis. Surgical resection is associated with significant morbidity and a significant risk of recurrence.1,2 Hill et al reported a 47% recurrence rate even after radical resection; however, they felt that excision was incomplete in 6 of the 7 recurrences.2 Despite the risks, surgical resection is often required for definitive treatment and results in symptomatic improvement in most patients. In this case, injection sclerotherapy was performed 4 times, but this treatment did not effectively treat the malformation and was associated with skin necrosis. After 4 treatments, it was obvious that the lesion was not going to respond to nonsurgical treatments and radical resection would be necessary. The treatment of malformations, whether surgical or nonsurgical, should be approached cautiously and treatment planned in consultation with interventional radiology colleagues. Upton et al in a review of 270 patients with vascular malformations, advocated a systematic, staged approach to resection from distal to proximal and dorsal to volar, awaiting return of function and softening of tissues prior to reoperation.1 They recommended complete excision of a single anatomic area at each stage. Repeat operations and postoperative immobilization can lead to scarring and stiffness. Given the importance of the thumb in hand function and our confidence in radical resection, immediate reconstruction was felt to be appropriate in this case. Yoshii et al5 first reported that radical resection of vascular malformations of the forearm and hand followed by flap coverage seemed to prevent recurrence, and this has been subsequently confirmed by others.9,11 Koshima et al7 resected a recurrent AVM of the index finger that was reconstructed with an extended wraparound flap, consisting of a wraparound flap combined with a dorsalis pedis flap. Adani et al8 reported resection of a low-flow VM of the distal phalanx of the thumb and reconstruction with a free toe pulp flap. Hattori et al10 also used a wraparound flap combined with a dorsalis pedis flap to cover the tendon and bony skeleton of the thumb after resection of an extensive AVM involving the thumb, index finger, and first web space. Toh et al11
reported 3 patients with AVMs involving the palmar aspect of the index finger, distal pulp of the thumb, and thenar eminence treated by radical resection and free flap coverage using 2 dorsalis pedis flaps and 1 peroneal flap. One patient had an ulcerating distal phalanx of the thumb that was reconstructed with a wraparound flap. Lu et al12 described resection of an AVM involving the thumb, thenar eminence, and first web space, and coverage of the tendon and bony skeleton of the thumb with a wraparound flap and the first web space covered with the extended dorsalis pedis flap. Of these 4 reported cases of VMs involving the thumb, the resultant defects after radical resection were relatively small in 2 patients, involving only the distal phalanx of the thumb and requiring reconstruction with a free toe pulp flap8 and a wraparound flap.11 Even the 2 patients with more extensive defects involving the thumb still had an intact bony skeleton and flexor and extensor tendons and required only soft tissue reconstruction with an extended wraparound flap.10,12 None of these 4 malformations approached the size of the VM presented here, which was truly massive and not only prevented pinch and grip function of the hand but also compromised elbow and shoulder function because of its weight. Additionally, this is a unique case of VM requiring complete amputation of the thumb and immediate reconstruction with an entire toe-to-thumb transfer. Toe-to-hand transfer has become a widely accepted technique for reconstruction of traumatic amputation of the digits since first described by Cobbett and Buncke.15–17 More recently, the indications for toe-to-hand transfers in children are evolving,18 –27 and toe transfers may become more preferable to other alternative techniques such as index finger pollicization, nonvascularized toe phalangeal bone grafting, and distraction lengthening.22 Timing of toe-to-hand transfers in children with congenital hand deformities is an important consideration. Most advocate transfer before age 4,18,23,24 with some recommending surgery around age 2,22,26,27 when the structures are large enough to perform the surgery without too much difficulty. In our case, the futility of nonsurgical management of the malformation became apparent during this optimal time period for toe-to-hand transfer. This case represents successful radical resection of a massive VM requiring amputation of the thumb and immediate reconstruction with a microsurgical second toe transfer. The child has excellent function and no evidence of recurrence after 5 years (Fig. 5).
Carlsen and Jones / Toe Transfer for Venous Malformation Received for publication March 27, 2007; accepted in revised form June 5, 2007. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Corresponding author: Neil F. Jones, MD, UCLA Hand Center, 10945 LeConte Ave., Suite 3355, Los Angeles, CA 90095; e-mail: njones@ mednet.ucla.edu. Copyright © 2007 by the American Society for Surgery of the Hand 0363-5023/07/32A10-0014$32.00/0 doi:10.1016/j.jhsa.2007.06.020
References 1. Upton J, Coombs CJ, Mulliken JB, Burrows PE, Pap S. Vascular malformations of the upper limb: A review of 270 patients. J Hand Surg 1999;24A:1019 –1035. 2. Hill RA, Pho RWH, Kumar VP. Resection of vascular malformations. J Hand Surg 1993;18B:17–21. 3. Szilagy DE, Smith RF, Elliott JP, Hageman JH, Congenital arteriovenous anomalies of the limbs. Arch Surg 1976;111: 423– 429. 4. Bogumill GP, Sullivan DJ, Baker GI. Tumors of the hand. Clin Orthop 1975;108:214 –222. 5. Yoshii T, Tamai S, Fukui N. Microsurgical approach for the hemangiomas and congenital arteriovenous fistulae in forearm and hand. J Jpn Soc Surg Hand 1985;2:736 –741. 6. Upton J, Mulliken JB, Murray JE. Classification and rationale for management of vascular anomalies in the upper extremity. J Hand Surg 1985;10A:970 –975. 7. Koshima I, Soeda S, Murashita T. Extended wrap-around flap for reconstruction of the finger with recurrent arteriovenous malformation. Plas Recon Surg 1993;91:1140 –1144. 8. Adani R, Busa R, Caroli A. Microsurgical approach for a venous malformation of the thumb. J Hand Surg 1997;22B: 30 –31. 9. Yamamoto Y, Ohura T, Minakawa H, Sugihara T, Yoshida T, Nohira K, Shintomi Y. Experience with arteriovenous malformations treated with flap coverage. Plas Recon Surg 1994;94(3):476 – 482. 10. Hattori Y, Doi K, Kawakami F, Watanabe M. Extended wrap-around flap for thumb reconstruction following radical excision of a congenital arteriovenous fistula. J Hand Surg 1998;23B:72–75. 11. Toh S, Tsubo K, Arai H, Harata S. Vascularized free flaps for reconstruction after resection of congenital arteriovenous malformations of the hand. J Recon Microsurg 2000;16(7); 511–517.
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12. Lu LI, Chen DJ, Chen HC, Coessens B. Arteriovenous malformation involving the thumb and hand: radical excision and reconstruction of multiple components. Ann Plast Surg 2002;49:414 – 418. 13. Mulliken JB Glowacki J. Hemangiomas and vascular malformations in infants and children: a classification based on endothelial characteristics. Plast Reconstr Surg 1982;69(3): 412– 422. 14. Vilanova JC, Barcelo J, Villalon M. MR and MR angiography characterization of soft tissue vascular malformations. Curr Probl Diagn Radiol 2004;33(4):161–170. 15. Cobbett JR. Free digital transfer: Report of a case of transfer of a great toe to replace an amputated thumb. J Bone Joint Surg 1969;51B:677– 679. 16. Buncke HJ, McLean DH, George PT, Creech BJ, Chater NL, Commons GW. Thumb replacement: Great toe transplantation by microvascular anastomosis. Br J Plast Surg 1973;26: 194 –201. 17. Morrison WA, O’Brien BM, MacLeod AM. Thumb reconstruction with a free neurovascular wrap-around flap from the big toe. J Hand Surg 1980;5:575–583. 18. Gilbert A. Toe transfers for congenital hand defects. J Hand Surg 1982;7:118 –124. 19. Gilbert A. Reconstruction of congenital hand defects with microvascular toe transfers. Hand Clin 1985;1:351–360. 20. Lister G. Microsurgical transfer of the second toe for congenital deficiency of the thumb. Plas Recon Surg 1988;82: 658 – 665. 21. Buck-Gramcko D. Progress in the treatment of congenital malformations of the hand. World J Surg 1990;14:715–724. 22. Vilkki SK. Advances in microsurgical reconstruction of the congenitally adactylous hand. Clin Orthop 1995;314:45–58. 23. Kay SP Wiberg M. Toe to hand transfer in children, part 1: technical aspects. J Hand Surg 1996;21B:723–734. 24. Kay SP, Wiberg M, Bellew M, Webb F. Toe to hand transfer in children, part 2: functional and psychological aspects. J Hand Surg 1996;21B:735–745. 25. Kay S McGuiness C. Microsurgical reconstruction in abnormalities of children’s hands. Hand Clin 1999;15:563–583. 26. Jones NF, Hansen SL, Bates SJ. Toe-to-hand transfers for congenital anomalies of the hand. Hand Clin 2007;23(1): 129. 27. Chang J Jones NF. Radiographic analysis of growth in pediatric microsurgical toe-to-hand transfers. Plas Recon Surg 2002;109:576 –582.