Embolotherapy in the treatment of congenital arteriovenous malformations of the hand: A case report

Embolotherapy in the treatment of congenital arteriovenous malformations of the hand: A case report

Vol. lOA, No. I January 1985 9 . Green 0: True and false traumatic aneurysms o f the hand. J Bone Joint Surg [Am]58: 120-8, 1973 10. Conn J, Bergan J...

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Vol. lOA, No. I January 1985

9 . Green 0: True and false traumatic aneurysms o f the hand. J Bone Joint Surg [Am]58: 120-8, 1973 10. Conn J, Bergan JJ , Bell JL: Hypothenar hammer syndrome: Post traumatic digital ischemia. Surgery 68: 1122-8, 1970 I I . Narsete EM: Traumatic aneurysm of the radial artery. Am J Surg 108:424-7, 1964 12 . Aulicino PL, Hutton PM], DuPuy TE: True palmar aneurysms-A case r eport and literature review. J HA ND SURG 7:613-6, 1982 13 . Layman CD , Ogden LL, Lister GO: True aneurys m of digital artery. J HAND SURG 7:617-8,1982 14 . May JW, Gross man JAI, Costas B: Cyanotic painful index and long fin gers associated with an asymptomatic ulnar artery aneurysms . J HAND S URG 7:622-5, 1982 15 . Rotenstein 0 , Gibas 0, Majmudar B , Chastain E: Famil-

ArteriTis WiTh aneurysms wiThin anatomic

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ial granulomatous arteritis with polyarthritis of juvenile onset. N Engl J Med 306:86-9, 1982 Lie JT: Disseminated visceral giant cell arteritisHistopathological description and differentiation from other granulomatous vasculitides . Am J Clin Pathol 69:299-304, 1978 Klein R, Hunder G, Stanson A, Sheps S: Large artery involvement in giant cell (temporal) arteritis. Ann Intern Med 83:806-12 , 1975 Miller R, Ferguson E: Temporal arteritis and polymyalgi a rheumatica- Two aspects of one disease . Postgrad Med 66:177-82 , 1979 Malmvall B, Bengtsson B, Alestig K, Bojs G, Iwarson S: The clinical pictures of giant cell arteritis . Pos tgrad Med 67:141 -8, 1980

Embolotherapy in the treatment of congenital arteriovenous malformations of the hand: A case report This article describes the treatment of a painful ulcerating congenital arteriovenous malformation in the hand of a 39-year-old woman. Preoperative transcatheter embolotherapy was used to diminish vascularity and engorgement. Operative treatment consisted of a resection of angiomatous material and coverage with a pedicle flap. Preoperative transcatheter embolization of small feeder vessels may offer some advantage over the operative approach alone in treating congenital arteriovenous malformations of the hand. (J HAND SURG 10A:135-9, 1985.)

1. Russell Moore , M.D., and Andrew 1. Weiland, M .D., Baltimore , Md .

Congenital arteriovenous malformations (CA VMs) of the upper extremity remain one of the

most challenging problems that doctors must treat. Short of ablation, operative treatment regimens frequently fail in cases that do not have discretely localized lesions. We describe a combined approach to

arteriovenous malformations (AVMs) of the hand that uses preoperative transcatheter embolization of the large feeder vessels, which is followed by partial resection of the arteriovenous (AV) fi stulas and coverage by a pedicle flap .

Case report From the Johns Hopkins University School of Medicine, Department of Orthopaedic Surgery , Baltimore, Md . Received for publication March 26, 1984; accepted in revised form May 9, 1984. Reprint requests: J. Russell Moore , M.D., Assistant Professor of Orthopaedic Surgery , The Good Samaritan Professional Bldg ., 5601 Loch Raven Blvd ., Baltimore, MD 21239.

A 39-year-old right-handed woman with rheumatoid arthritis was referred for evaluation of a painful, stiff, and ulcerated left thumb and index finger (Fig. 1). She had been diagnosed as having a CA VM of the left upper extremity as a child and had undergone several ligations of the brachial artery to diminish the deformity. The patient reported progressive pain and dysfunction of her left index finger and thumb ,

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Digital subtraction angiography and embolization of the CA VM were performed by cannulating the left radial artery with an 18-gauge long-dwell catheter. A solution of isobutyl 2-cyanoacrylate (ISe) was injected; it successfully blocked the malformation at the base of the left thumb and also occluded the draining veins in the interosseous membrane (Fig. 2, B and C). The radial artery was ligated at the end of the procedure, and vascular engorgement of the hand was greatly diminished. Two days later, the patient underwent a ray resection of the index finger, skeletonization of the ulnar border of the thumb, and primary pedicle flap coverage with a neurovascular island flap from the ulnar aspect of the ring finger (Fig. 3). The thumb tip became devascularized as a result of the surgical resection, and the distal phalanx and nail were subsequently excised. Six months later at follow-up, the patient was without pain, had no recurrence of ulceration, and had an excellent cosmetic result (Fig. 4), although sensation in the thumb was still perceived as emanating from the ring finger. Most important, she was able to use her hand for the first time in more than a year.

Discussion

Fig. I, A-B. The appearance of the left hand before embolotherapy. The index finger and thumb are stiff, engorged, and dysesthetic. and was so frustrated over the recurrent ulceration of these digits that she would have considered amputation to resolve her problem. She was a nonsmoker. A physical examination failed to disclose any evidence of cardiomegaly or changes secondary to a high-output state. Pertinent features of the physical examination were related to the left upper extremity. MUltiple surgical scars secondary to proximal vessel ligation were noted. There were no bruits or thrills noted in the left upper extremity. However, a prominent subclavian pulsation was visible in the supraclavicular fossa. There was no enlargement or cyanosis of the forearm except for varicose veins of the forearm and hand. The thumb and index finger were discolored, stiff in extension, and dysesthetic. In addition, the patient had an ulcer at the base of the thumb that would not heal. All other digits were normal. The patient had a weak radial pulse; however, the ulnar pulse was strong. An Allen test showed sufficient collateral circulation to supply the hand when either the radial or ulnar artery was occluded. An angiogram showed an absence of the brachial artery with the axillary artery supplying the arm via numerous collaterals that rejoined at the elbow and reformed as the interosseous and very large ulnar artery. Several small branches of the radial artery were present and fed a large AVM of the thumb and index finger (Fig. 2, A).

CAVMs result from a failure in differentiation of the common embryonic anlage into a true artery and vein and persistent communication between them.I-:1 These developmental communications may become active shunts either spontaneously or as a result of trauma. The course of treatment mainly depends on the extent of the AVM and the severity of symptoms as described by the patient. A physical examination should include proximal search for AVMs and an evaluation of the cardiovascular system as a whole. Although it is unlikely that AVMs of the hand cause any systemic effects, larger proximal shunts can cause significant hemodynamic alterations including cardiac hypertrophy and increased cardiac output. 4 Proximal compression of a feeding vessel may decrease the heart rate (Branham's sign) in large fistulas.:;' 6 The Doppler ultrasonic flow detector may be sensitive enough to locate small AVMs and is a useful adjunct to the physical examination. Plain radiographs are useful for detecting soft tissue masses as well as phleboliths and bony erosion. An arteriogram is mandatory and enables one to accurately locate the AV communications while simultaneously visualizing vital collateral channels that are necessary to ensure survival of essential parts, 1, 7 Treatment of CAVMs has been disappointing, Complete surgical excision is the procedure of choice for a discrete AVM. However more extensive and diffuse AVMs are usually encountered. I, H, P Proximal vessel ligation alone has proved unsuccessful because of subsequent extensive development of collaterals, 10. II

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Fig. 2. A, An arteriogram of the hand reveals a large AVM involving the thumb and index finger that is being fed by several small branches of the radial artery. B, An angiogram of the hand after two injections of me. C, The lIrrow indicates the location of the thrombotic substances on the radiograph of the hand after injection.

However, the results of ligation combined with excision of the fistula may occasionally be satisfactory.8 Embolotherapy was initially viewed as an emergency procedure of desperation l2 ; however, technical improvements in catheters, thrombotic substances, and imaging techniques have made it possible for interventional radiologists to safely occlude hard-to-find feeder vessels by transcatheter embolization .D. 1:1-1;; The most important use of transcatheter embolization is in treating AVMs in inaccessible areas of the body such as the liver, retroperitoneal space , and intracranial sites, and transcatheter embolization may be used preoperatively to diminish blood loss at the time of an operative resection. 9 . 1:1. 16 It is frequently difficult during surgery of the hand to visually identify and ligate the main tributaries leading to an AVM . The interventional radiologist can perform embolization very safely in conjunction with a preoperative arteriogram of the hand . Potential complications include ischemia and ischemic pain after embolization, 17 an inflammatory reaction to thrombotic substances,lx. 19 and recanalization of vessels .20 Various materials have been used for embolotherapy

and include autologous blood , Gelfoam, polyvinyl alcohol foam (lvalon), oxidized cellulose (Oxycel), microfibrillar collagen (Avatine), wool and steel coils, and silicone balloons. 21 We have described the use of mc (bucrylate).2o IBC is a liquid tissue adhesive that polymerizes when it comes into contact with blood , and the resulting thrombus produces long-term occlusion . mc remains an investigational drug and is not yet available for general use. In sites other than the hand, curative embolization of AVMs with IBC and silicone rubber has been reported .22 - 2:J Although it is unlikely that transcatheter embolization will be the sole invasive treatment for a CA VM, it is a useful adjunct to surgery. Embolotherapy may supplant operative ligation of tributary vessels to AVMs because of accuracy , speed, and ease of placement of a suitable thrombus and the decrease in surgical exposure that is needed during definitive resection of an AVM. Recurrent ulceration and pain in a digit usually will not abate unless a significant portion of the AVM is excised. I A definitive resection of an AVM in the hand often requires that digits be amputated or that subtotal

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Fig. 3. A, Stippled areas (thumb and index finger) on a preoperative photograph indicate the areas of resectio n and donor skin from the ring finger. B, The appearance of the hand after ray re section of the index finger, coverage with pedicle fl ap , and grafting of skin.

Fig. 4, A-B. The appearance of the hand at follow-up 6 months after surgery . The patient had an interim procedure to remove the nail and shorten the di stal phalanx. Pain was relieved , cosme sis was improved , and the patient has a functioning hand.

removal of soft tissue and skin into which the angiomatous tissue has grown be performed. 26- 28 One should always be cautious in claiming a cure for AVMs , especially if treatment other than amputation has been performed. Recurrence and continued proliferation are well-recognized sequelae. Preoperative em-

bolotherapy may facilitate the dissection and removal of proliferative AV fistulous masses . The application of interventional radiology in treating vascular disorders of the hand is in its infancy; however, it seems a natural extension of the rapidly expanding technology of radiologic imaging techniques.

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