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False aneurysm and brachial plexus palsy complicating a proximal humeral exostosis
FALSE A N E U R Y S M A N D B R A C H I A L P L E X U S PALSY COMPLICATING A PROXIMAL HUMERAL EXOSTOSIS C. H. GERRAND
From the Department of Orthopaedics, Western Infirmary, Glasgow, UK An unusual case is described in which a false aneurysm of the brachial artery secondary to an exostosis of the proximal humerus caused a compressive lesion of the brachial plexus. Surgical treatment of the exostosis and the false aneurysm relieved the symptoms.
Journal of Hand Surgery (British and European Volume, 1997) 22B: 3:413-415 False aneurysms can develop where a vessel has been damaged by an adjacent exostosis. This has been reported most often for the popliteal artery (Brailsford, 1948; Cassie et al, 1975; Clark and Keokarn, 1965; Denman et al, 1959; Hasselgren et al, 1983; Hershey and Lansden, 1972; Hovelius, 1975; Hudson, 1955; Lesser and Greeley, 1958; Masson and Pullan, 1966; Mukerjea, 1967; Paul, 1953; Shah, 1978; Vallance et al, 1985). Complications involving the brachial artery are less common (Cachera et al, 1970; Nevelsteen et al, 1988; Vallance et al, 1985). Radial and axillary nerve palsies have been reported secondary to proximal humeral exostoses (Coenen and Biltjes, 1992; Witthaut et al, 1994), but lesions of the brachial plexus have not. This is a report of a false aneurysm arising secondary to a proximal humeral exostosis which caused a compressive lesion of the infraclavicular brachial plexus in addition to a radial nerve palsy caused by the exostosis alone.
compressed by the aneurysm rather than the exostosis. The artery was repaired with 610 nylon and the upper part of the exostosis was removed. The remainder arose more distally and posteriorly and could not be resected easily through this incision. However, it did not interfere
CASE R E P O R T
A 16-year-old boy had two episodes of temporary paralysis of his left arm within a week. The first occurred after weight training, a sport which he had recently taken up, and the second after throwing a football during a match. During both activities the shoulder was held flexed and abducted. Considerable bruising developed in the axilla. An axillary swelling which he had had all his life had recently enlarged. On examination there was a large mass, anteromedial on the upper arm which prevented complete adduction of the limb. He had slight weakness of the extensors of his wrist and fingers, but his paralysis had otherwise recovered. X-rays showed an exostosis on the medial aspect of the proximal humerus, 9 cm in length and shaped like the keel of a boat (Fig 1). Two weeks later, he presented with global weakness of the upper limb, most marked in the flexor and extensor muscles of the elbow, wrist and fingers, and the intrinsic muscles of the hand. The mass on the upper arm had enlarged and was pulsatile with an overlying bruit. Angiography confirmed the presence of a false aneurysm, 6 cm in diameter, at the level of the proximal end of the exostosis (Fig 2). At operation, through a deltopectoral approach, the aneurysm was opened and a defect in the brachial artery was found adjacent to the sharp medial edge of the exostosis. The infraclavicular brachial plexus had been 413
Fig 1
X-ray showing an exostosis of the proximal humerus.
Fig 2
An angiogram showing a false aneurysm arising adjacent to the exostosis.
THE JOURNAL OF HAND SURGERY VOL. 22B No. 3 JUNE 1997
414
with the neurovascular bundle and was therefore not removed. Over the next month function returned to the finger flexors and the intrinsic muscles of the hand, but the radial nerve palsy persisted. Eight months after the first procedure there was active contraction of the triceps but persistent weakness of the wrist and finger extensors. The remaining part of the exostosis had enlarged clinically. A second procedure was performed through a posterior approach to remove the more distal part of the exostosis which was found lying close to the musculospiral groove with the radial nerve stretched over it. Two months later the radial nerve palsy recovered. One year later there was no clinical or radiological evidence of recurrence (Fig 3). Both tumours were benign osteocartilaginous exostoses on histological examination. DISCUSSION It is well recognized that an exostosis which lies next to a vessel can cause vascular complications. Although Cachera et al (1970) and Nevelsteen et al (1988) state this was first described by Paul (1953), there is an earlier report by Brailsford (1948). The most common complication is a false aneurysm (Brailsford, 1948; Cachera et al, 1970; Cassie et al, 1975; Clark and Keokarn, 1965; Denman et al, 1959; Hasselgren et al, 1983; Hershey and Lansden, 1972; Hovelius, 1975; Hudson, 1955; Masson and Pullan, 1966; Mukerjea, 1967; Paul, 1953; Shah, 1978; Vallance et al, 1985) although an arteriovenous fistula has been described (Lesser and Greeley, 1958). Damage to vessels tends to occur at skeletal maturity when the cartilaginous cap of the exostosis ossifies, and may cause repeated minor trauma (Hershey and Lansden, 1972), acute laceration (Mukerjea, 1967) or avulsion of an arterial branch (Cassie et al, 1975). The exostosis may be solitary (Hershey and Lansden, 1972; Shah, 1978) or occur in association with hereditary multiple exostoses (Clark and Keokarn, 1965; Denman et al, 1959; Hershey and Lansden, 1972; Hudson, 1955; Masson and Pullan, 1966; Mukerjea, 1967; Paul, 1953; Vallance et al, 1985). Pain and swelling may follow exertion (Cachera et al, 1970) or a single traumatic episode (Mukeljea, 1967). There may be symptoms of vascular insufficiency (Lesser and Greeley, 1958; Masson and Pullan, 1966; Shah, 1978; Vallance et al, 1985). Clinical examination may reveal a large swelling, occasionally pulsatile, with evidence of impaired distal perfusion (Hershey and Lansden, 1972; Hovelius, 1975; Paul, 1953). There may be a bruit overlying the mass (Hershey and Lansden, 1972; Mukerjea, 1967) and restricted movement of the adjacent joint (Clark and Keokarn, 1965; Hovelius, 1975; Mukerjea, 1967). The presentation may mimic that of malignant change in an exostosis (Brailsford, 1948; Hasselgren et al, 1983; Hovelius, 1975; Hudson, 1955; Mukerjea, 1967).
Fig 3
X-ray 1 year after surgery shows no evidence of recurrence of the exostosis.
Preoperative angiography is recommended if vascular involvement is suspected (Vallance et al, 1985). Vascular compromise may demand urgent surgery (Hudson, 1955). An arterial perforation may be closed primarily if it is small (Mukerjea, 1967; Shah, 1978), as in this case, or may require resection and end-to-end anastomosis (Denman et al, 1959; Hershey and Lansden, 1972; Nevelsteen et al, 1988), or interposition graft (Hasselgren et al, 1983; Masson and Pullan, 1966). The exostosis should be removed at the same procedure (Hasselgren et al, 1983). Neurological complications of proximal humeral exostoses are uncommon. Axillary nerve and radial nerve entrapment have been described, both of which recovered after resection of the exostosis (Coenen and Biltjes, 1992; Witthaut et al, 1994). A false aneurysm caused by an exostosis can itself cause neurological complications which may be overlooked. In this case, it was
COMPLICATIONS OF H U M E R A L EXOSTOSIS
clear at operation that the aneurysm rather than the exostosis was compressing the brachial plexus. This was quite distinct from the radial nerve compression which arose from a more distal part of the exostosis. It is suggested that when an aneurysm arises adjacent to an exostosis either may produce neurological symptoms and both should be adequately treated. The value of prophylactic excision of an exostosis on these grounds alone has been disputed (Denman et al, 1959; Hershey and Lansden, 1972; Masson and Pullan, 1966; Mukerjea, 1967; Shah, 1978) but is probably not justified, as the results of surgery for vascular complications are good (Nevelsteen et al, 1988).
Acknowledgements Many thanks to M r J. F. Crossan and Mr R Rogers for allowing me to report their patient.
References Brailsford JF. The radiology of bones andjoints, 4th edn. London, J&A Churchill, 1948: 215. Cachera JP, Letournel E, Kieffer E (1970). A case of arterial aneurysm complicating an osteogenic exostosis of the upper extremity of the humerus. [French]. Journal de Chirurgie, 99: 3 9 4 4 . Cassie GF, Dawson AS, Sheville E (1975). False aneurysm of femoral artery from cancellous exostosis of femur. Report of a case in a boy of fourteen. Journal of Bone and Joint Surgery, 57B: 379. Clark PM, Keokarn T (t965). Popliteal aneurysm complicating benign osteocartilaginous exostosis. Journal of Bone and Joint Surgery, 47A: 1386-1388. Coenen L, Biltjes I (1992). High radial nerve palsy caused by a humeral exostosis: a case report. Journal of H a n d Surgery, 17A: 668 669.
415 Denman FR, Shindler TO, H a m p t o n J, Hanson L (1959). Aneurysm of the popliteal artery caused by osteochondroma of the femur. Journal of Bone and Joint Surgery, 41A: 1526-1528. Hasselgren PO, Eriksson B, Lukes P, Seeman T (1983). False popliteal aneurysm caused by exostosis of the femur. Journal of Cardiovascular Surgery, 24: 54~542. Hershey SL, Lansden FT (1972). Osteochondromas as a cause of false popliteal aneurysms. Review of the literature and report of two cases. Journal of Bone and Joint Surgery, 54A: t765 1768. Hovelius L (1975). Aneurysm of popliteal artery caused by cartilaginous exostosis. A case report. Acta Orthopaedica Scandinavica, 46: 836-838. Hudson OC (1955). Traumatic aneurysm of the popliteal artery due to osteochondroma. American Journal of Surgery, 90: 528-530. Lesser AJ, Greeley CE (1958). Femoropopliteal arteriovenous aneurysm caused by fractured osteochondroma of the femur. Journal of the American Medical Association, 167: 1830-1833. Masson AF, Pullan JM (1966). Aneurysm complicating exostosis. British Journal of Surgery, 53: 929-932. Mukerjea SK (1967). Traumatic aneurysm of the popliteal artery due to osteochondroma. British Journal of Surgery, 54:810-811. Nevelsteen A, Pype P, Broos P, Suy R (1988). Brachial artery rupture due to an exostosis: brief report. Journal of Bone and Joint Surgery, 70B: 672. Paul M (1953). Aneurysm of the popliteal artery from perforation by a cancellous exostosis of the fenmr. Journal of Bone and Joint Surgery, 35B: 27C~271. Shah PJ (1978). Aneurysm of the popliteal artery secondary to trauma from an osteochondroma of the femur: a case report and review of the literature. British Journal of Surgery, 65: 786-788. Vallance R, Hamblen DL, Kelly IG (1985). Vascular complications of osteochondroma. Clinical Radiology, 36:639 642. Witthaut J, Steffens K J, Koob E (1994). Intermittent axillary nerve palsy caused by a humeral exostosis. Journal of H a n d Surgery, 19B: 4 2 2 4 2 3 .
Received: 3 October 1996 Accepted after revision: 30 December 1996 Mr C.H. Gerrand, Department of Orthopaedics, Western Infirmary, Dumbarton Road, Glasgow GI 1 6NT, UK. © 1997The British Society for Surgery of the Hand
From the Department of Orthopaedics, Western Infirmary, Glasgow, UK An unusual case is described in which a false aneurysm of the brachial artery secondary to an exostosis of the proximal humerus caused a compressive lesion of the brachial plexus. Surgical treatment of the exostosis and the false aneurysm relieved the symptoms.
Journal of Hand Surgery (British and European Volume, 1997) 22B: 3:413-415 False aneurysms can develop where a vessel has been damaged by an adjacent exostosis. This has been reported most often for the popliteal artery (Brailsford, 1948; Cassie et al, 1975; Clark and Keokarn, 1965; Denman et al, 1959; Hasselgren et al, 1983; Hershey and Lansden, 1972; Hovelius, 1975; Hudson, 1955; Lesser and Greeley, 1958; Masson and Pullan, 1966; Mukerjea, 1967; Paul, 1953; Shah, 1978; Vallance et al, 1985). Complications involving the brachial artery are less common (Cachera et al, 1970; Nevelsteen et al, 1988; Vallance et al, 1985). Radial and axillary nerve palsies have been reported secondary to proximal humeral exostoses (Coenen and Biltjes, 1992; Witthaut et al, 1994), but lesions of the brachial plexus have not. This is a report of a false aneurysm arising secondary to a proximal humeral exostosis which caused a compressive lesion of the infraclavicular brachial plexus in addition to a radial nerve palsy caused by the exostosis alone.
compressed by the aneurysm rather than the exostosis. The artery was repaired with 610 nylon and the upper part of the exostosis was removed. The remainder arose more distally and posteriorly and could not be resected easily through this incision. However, it did not interfere
CASE R E P O R T
A 16-year-old boy had two episodes of temporary paralysis of his left arm within a week. The first occurred after weight training, a sport which he had recently taken up, and the second after throwing a football during a match. During both activities the shoulder was held flexed and abducted. Considerable bruising developed in the axilla. An axillary swelling which he had had all his life had recently enlarged. On examination there was a large mass, anteromedial on the upper arm which prevented complete adduction of the limb. He had slight weakness of the extensors of his wrist and fingers, but his paralysis had otherwise recovered. X-rays showed an exostosis on the medial aspect of the proximal humerus, 9 cm in length and shaped like the keel of a boat (Fig 1). Two weeks later, he presented with global weakness of the upper limb, most marked in the flexor and extensor muscles of the elbow, wrist and fingers, and the intrinsic muscles of the hand. The mass on the upper arm had enlarged and was pulsatile with an overlying bruit. Angiography confirmed the presence of a false aneurysm, 6 cm in diameter, at the level of the proximal end of the exostosis (Fig 2). At operation, through a deltopectoral approach, the aneurysm was opened and a defect in the brachial artery was found adjacent to the sharp medial edge of the exostosis. The infraclavicular brachial plexus had been 413
Fig 1
X-ray showing an exostosis of the proximal humerus.
Fig 2
An angiogram showing a false aneurysm arising adjacent to the exostosis.
THE JOURNAL OF HAND SURGERY VOL. 22B No. 3 JUNE 1997
414
with the neurovascular bundle and was therefore not removed. Over the next month function returned to the finger flexors and the intrinsic muscles of the hand, but the radial nerve palsy persisted. Eight months after the first procedure there was active contraction of the triceps but persistent weakness of the wrist and finger extensors. The remaining part of the exostosis had enlarged clinically. A second procedure was performed through a posterior approach to remove the more distal part of the exostosis which was found lying close to the musculospiral groove with the radial nerve stretched over it. Two months later the radial nerve palsy recovered. One year later there was no clinical or radiological evidence of recurrence (Fig 3). Both tumours were benign osteocartilaginous exostoses on histological examination. DISCUSSION It is well recognized that an exostosis which lies next to a vessel can cause vascular complications. Although Cachera et al (1970) and Nevelsteen et al (1988) state this was first described by Paul (1953), there is an earlier report by Brailsford (1948). The most common complication is a false aneurysm (Brailsford, 1948; Cachera et al, 1970; Cassie et al, 1975; Clark and Keokarn, 1965; Denman et al, 1959; Hasselgren et al, 1983; Hershey and Lansden, 1972; Hovelius, 1975; Hudson, 1955; Masson and Pullan, 1966; Mukerjea, 1967; Paul, 1953; Shah, 1978; Vallance et al, 1985) although an arteriovenous fistula has been described (Lesser and Greeley, 1958). Damage to vessels tends to occur at skeletal maturity when the cartilaginous cap of the exostosis ossifies, and may cause repeated minor trauma (Hershey and Lansden, 1972), acute laceration (Mukerjea, 1967) or avulsion of an arterial branch (Cassie et al, 1975). The exostosis may be solitary (Hershey and Lansden, 1972; Shah, 1978) or occur in association with hereditary multiple exostoses (Clark and Keokarn, 1965; Denman et al, 1959; Hershey and Lansden, 1972; Hudson, 1955; Masson and Pullan, 1966; Mukerjea, 1967; Paul, 1953; Vallance et al, 1985). Pain and swelling may follow exertion (Cachera et al, 1970) or a single traumatic episode (Mukeljea, 1967). There may be symptoms of vascular insufficiency (Lesser and Greeley, 1958; Masson and Pullan, 1966; Shah, 1978; Vallance et al, 1985). Clinical examination may reveal a large swelling, occasionally pulsatile, with evidence of impaired distal perfusion (Hershey and Lansden, 1972; Hovelius, 1975; Paul, 1953). There may be a bruit overlying the mass (Hershey and Lansden, 1972; Mukerjea, 1967) and restricted movement of the adjacent joint (Clark and Keokarn, 1965; Hovelius, 1975; Mukerjea, 1967). The presentation may mimic that of malignant change in an exostosis (Brailsford, 1948; Hasselgren et al, 1983; Hovelius, 1975; Hudson, 1955; Mukerjea, 1967).
Fig 3
X-ray 1 year after surgery shows no evidence of recurrence of the exostosis.
Preoperative angiography is recommended if vascular involvement is suspected (Vallance et al, 1985). Vascular compromise may demand urgent surgery (Hudson, 1955). An arterial perforation may be closed primarily if it is small (Mukerjea, 1967; Shah, 1978), as in this case, or may require resection and end-to-end anastomosis (Denman et al, 1959; Hershey and Lansden, 1972; Nevelsteen et al, 1988), or interposition graft (Hasselgren et al, 1983; Masson and Pullan, 1966). The exostosis should be removed at the same procedure (Hasselgren et al, 1983). Neurological complications of proximal humeral exostoses are uncommon. Axillary nerve and radial nerve entrapment have been described, both of which recovered after resection of the exostosis (Coenen and Biltjes, 1992; Witthaut et al, 1994). A false aneurysm caused by an exostosis can itself cause neurological complications which may be overlooked. In this case, it was
COMPLICATIONS OF H U M E R A L EXOSTOSIS
clear at operation that the aneurysm rather than the exostosis was compressing the brachial plexus. This was quite distinct from the radial nerve compression which arose from a more distal part of the exostosis. It is suggested that when an aneurysm arises adjacent to an exostosis either may produce neurological symptoms and both should be adequately treated. The value of prophylactic excision of an exostosis on these grounds alone has been disputed (Denman et al, 1959; Hershey and Lansden, 1972; Masson and Pullan, 1966; Mukerjea, 1967; Shah, 1978) but is probably not justified, as the results of surgery for vascular complications are good (Nevelsteen et al, 1988).
Acknowledgements Many thanks to M r J. F. Crossan and Mr R Rogers for allowing me to report their patient.
References Brailsford JF. The radiology of bones andjoints, 4th edn. London, J&A Churchill, 1948: 215. Cachera JP, Letournel E, Kieffer E (1970). A case of arterial aneurysm complicating an osteogenic exostosis of the upper extremity of the humerus. [French]. Journal de Chirurgie, 99: 3 9 4 4 . Cassie GF, Dawson AS, Sheville E (1975). False aneurysm of femoral artery from cancellous exostosis of femur. Report of a case in a boy of fourteen. Journal of Bone and Joint Surgery, 57B: 379. Clark PM, Keokarn T (t965). Popliteal aneurysm complicating benign osteocartilaginous exostosis. Journal of Bone and Joint Surgery, 47A: 1386-1388. Coenen L, Biltjes I (1992). High radial nerve palsy caused by a humeral exostosis: a case report. Journal of H a n d Surgery, 17A: 668 669.
415 Denman FR, Shindler TO, H a m p t o n J, Hanson L (1959). Aneurysm of the popliteal artery caused by osteochondroma of the femur. Journal of Bone and Joint Surgery, 41A: 1526-1528. Hasselgren PO, Eriksson B, Lukes P, Seeman T (1983). False popliteal aneurysm caused by exostosis of the femur. Journal of Cardiovascular Surgery, 24: 54~542. Hershey SL, Lansden FT (1972). Osteochondromas as a cause of false popliteal aneurysms. Review of the literature and report of two cases. Journal of Bone and Joint Surgery, 54A: t765 1768. Hovelius L (1975). Aneurysm of popliteal artery caused by cartilaginous exostosis. A case report. Acta Orthopaedica Scandinavica, 46: 836-838. Hudson OC (1955). Traumatic aneurysm of the popliteal artery due to osteochondroma. American Journal of Surgery, 90: 528-530. Lesser AJ, Greeley CE (1958). Femoropopliteal arteriovenous aneurysm caused by fractured osteochondroma of the femur. Journal of the American Medical Association, 167: 1830-1833. Masson AF, Pullan JM (1966). Aneurysm complicating exostosis. British Journal of Surgery, 53: 929-932. Mukerjea SK (1967). Traumatic aneurysm of the popliteal artery due to osteochondroma. British Journal of Surgery, 54:810-811. Nevelsteen A, Pype P, Broos P, Suy R (1988). Brachial artery rupture due to an exostosis: brief report. Journal of Bone and Joint Surgery, 70B: 672. Paul M (1953). Aneurysm of the popliteal artery from perforation by a cancellous exostosis of the fenmr. Journal of Bone and Joint Surgery, 35B: 27C~271. Shah PJ (1978). Aneurysm of the popliteal artery secondary to trauma from an osteochondroma of the femur: a case report and review of the literature. British Journal of Surgery, 65: 786-788. Vallance R, Hamblen DL, Kelly IG (1985). Vascular complications of osteochondroma. Clinical Radiology, 36:639 642. Witthaut J, Steffens K J, Koob E (1994). Intermittent axillary nerve palsy caused by a humeral exostosis. Journal of H a n d Surgery, 19B: 4 2 2 4 2 3 .
Received: 3 October 1996 Accepted after revision: 30 December 1996 Mr C.H. Gerrand, Department of Orthopaedics, Western Infirmary, Dumbarton Road, Glasgow GI 1 6NT, UK. © 1997The British Society for Surgery of the Hand