Portal de Periódicos da CAPES

True ulnar artery aneurysm of the hand in an 18-month-old boy: A case report

2007; Elsevier BV; Volume: 45; Issue: 4 Linguagem: Inglês

10.1016/j.jvs.2006.11.041

1097-6809

Mohammed Al‐Omran,

Cardiovascular Effects of Exercise

True aneurysms of the hand arteries are rare and are exceedingly uncommon in children. Presented is a case of a true ulnar artery aneurysm in an 18-month-old boy in which there was no history of trauma. The aneurysm was resected without reconstruction because of the normal preoperative Allen test result, normal preoperative finger pressure measurement with ulnar artery occlusion, the angiographic evidence that the radial artery was the dominant artery of the hand, and intraoperative evidence of adequate hand perfusion after excluding the aneurysm from the hand circulation as documented by good Doppler signals in all digital arteries. True aneurysms of the hand arteries are rare and are exceedingly uncommon in children. Presented is a case of a true ulnar artery aneurysm in an 18-month-old boy in which there was no history of trauma. The aneurysm was resected without reconstruction because of the normal preoperative Allen test result, normal preoperative finger pressure measurement with ulnar artery occlusion, the angiographic evidence that the radial artery was the dominant artery of the hand, and intraoperative evidence of adequate hand perfusion after excluding the aneurysm from the hand circulation as documented by good Doppler signals in all digital arteries. Distal ulnar artery aneurysms, although uncommon, have been well described in adults as a clinical finding as a part of the hypothenar hammer syndrome.1Ferris B.L. Taylor Jr, L.M. Oyama K. McLafferty R.B. Edwards J.M. Moneta G.L. et al.Hypothenar hammer syndrome: proposed etiology.J Vasc Surg. 2000; 31: 104-113Abstract Full Text Full Text PDF PubMed Scopus (149) Google Scholar, 2Cooke R.A. Hypothenar hammer syndrome: a discrete syndrome to be distinguished from hand-arm vibration syndrome.Occup Med (Lond). 2003; 53: 320-324Crossref PubMed Scopus (63) Google Scholar Congenital true aneurysms of the ulnar artery, however, are rare.3Offer G.J. Sully L. Congenital aneurysm of the ulnar artery in the palm.J Hand Surg (Br). 1999; 24: 735-737Crossref PubMed Scopus (23) Google Scholar This article presents a case of an ulnar artery true aneurysm presenting as a mass in the palm of a toddler and discusses the diagnostic evaluation and treatment. The case is discussed in the context of other reported cases of ulnar artery aneurysms in children.3Offer G.J. Sully L. Congenital aneurysm of the ulnar artery in the palm.J Hand Surg (Br). 1999; 24: 735-737Crossref PubMed Scopus (23) Google Scholar, 4Deune E.G. McCarthy E.F. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis.Orthopedics. 2005; 28: 1459-1461PubMed Google Scholar, 5Witt P.D. Bowen K.A. Johansen K. True ulnar artery aneurysm of the hand in an 8-year-old boy.Plast Reconstr Surg. 2003; 111: 2475-2476PubMed Google Scholar, 6Cron J. Saliou C. Fabiani J.N. Traumatic aneurysm of the ulnar artery in a child.Injury. 1997; 28: 401-403Abstract Full Text PDF PubMed Scopus (11) Google Scholar An 18-month-old boy presented with a pulsatile mass located in the inner aspect of the left hypothenar eminence. The mass was incidentally discovered by his pediatrician during a routine follow-up 3 months before the referral. During that time, the mass had enlarged, but no signs or symptoms were suggestive of a thromboembolic event. The patient was a product of a full-term, uncomplicated pregnancy, and his medical history was unremarkable. His parents reported no previous hand-penetrating injury or blunt trauma. His family history was unremarkable. On physical examination, the patient appeared healthy, without any dysmorphic features. The left hand examination showed a 1-cm pulsatile, nontender, compressible mass in the inner aspect of the hypothenar eminence. There was no thrill or bruit over the mass. The radial and ulnar arteries were palpable at the wrist, with a normal Allen test result. No signs of finger ischemia were observed. Results of a routine laboratory investigation of his blood, erythrocyte sedimentation rate, and C-reactive protein were within normal limits. The test result for antinuclear antibody was negative. An arterial duplex study of the left upper limb revealed an ulnar artery aneurysm distal to the wrist crease. The aneurysm was 1 cm in diameter and 3 cm in length, with a small mural thrombus. The left hand finger pressure measurement showed normal readings. No drop in the finger pressures was observed when the ulnar artery was occluded; however, a mild drop in the pressure was observed when the radial artery was occluded. An arteriogram of the left upper limb demonstrated a fusiform distal ulnar artery aneurysm in the palm and incomplete superficial and deep palmar arches (Fig 1). The digital arteries to the thumb and fingers were patent. No radiologic evidence of distal arterial embolization was found. A selective radial and ulnar arteriogram showed that the radial artery was the dominant artery of the hand (Fig 2).Fig 2Selective left radial artery arteriogram.View Large Image Figure ViewerDownload Hi-res image Download (PPT) During surgical exploration, a longitudinal skin incision was made directly over the aneurysm. Sharp dissection through the fascia revealed a 1.5-cm ulnar artery aneurysm. After identification of the ulnar nerve, dissection was done and vessel loops were used to control the aneurysm proximally and distally (Fig 3). A trial of intraoperative clamping of the aneurysm proximally and distally resulted in a satisfactory perfusion of the hand as documented by good Doppler signals in all digital arteries. All vessels that communicated with the aneurysm were therefore ligated and the aneurysm sac was resected. Histology demonstrated a true aneurysm where all three layers (intima, media, and adventitia) of the arterial wall were seen with hyperplastic intimal layer and fragmentation of elastin fibres in the media. No evidence of vasculitis was observed. The patient’s postoperative course was uneventful, and he was discharged home on the second postoperative day. At follow-up 7 months later, the patient had normal left hand function and good perfusion, as demonstrated by an arterial duplex study and normal digital pressures. Ulnar artery aneurysm in adults, although unusual, has been most commonly identified in conjunction with the hypothenar hammer syndrome, which is typically an occupationally acquired disease.1Ferris B.L. Taylor Jr, L.M. Oyama K. McLafferty R.B. Edwards J.M. Moneta G.L. et al.Hypothenar hammer syndrome: proposed etiology.J Vasc Surg. 2000; 31: 104-113Abstract Full Text Full Text PDF PubMed Scopus (149) Google Scholar, 2Cooke R.A. Hypothenar hammer syndrome: a discrete syndrome to be distinguished from hand-arm vibration syndrome.Occup Med (Lond). 2003; 53: 320-324Crossref PubMed Scopus (63) Google Scholar The etiology is thought to be due to repetitive trauma to the vulnerable portion of the ulnar artery as it courses around the hook of the hamate bone in the wrist.1Ferris B.L. Taylor Jr, L.M. Oyama K. McLafferty R.B. Edwards J.M. Moneta G.L. et al.Hypothenar hammer syndrome: proposed etiology.J Vasc Surg. 2000; 31: 104-113Abstract Full Text Full Text PDF PubMed Scopus (149) Google Scholar, 2Cooke R.A. Hypothenar hammer syndrome: a discrete syndrome to be distinguished from hand-arm vibration syndrome.Occup Med (Lond). 2003; 53: 320-324Crossref PubMed Scopus (63) Google Scholar In the pediatric population, however, the finding of an ulnar artery aneurysm is very rare. A computerized literature search was conducted in MEDLINE (1966 to September 2006) and EMBASE (1980 to September 2006). The search used the keywords ulnar artery and aneurysm. Four cases of ulnar artery aneurysm have been reported in the pediatric population3Offer G.J. Sully L. Congenital aneurysm of the ulnar artery in the palm.J Hand Surg (Br). 1999; 24: 735-737Crossref PubMed Scopus (23) Google Scholar, 4Deune E.G. McCarthy E.F. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis.Orthopedics. 2005; 28: 1459-1461PubMed Google Scholar, 5Witt P.D. Bowen K.A. Johansen K. True ulnar artery aneurysm of the hand in an 8-year-old boy.Plast Reconstr Surg. 2003; 111: 2475-2476PubMed Google Scholar, 6Cron J. Saliou C. Fabiani J.N. Traumatic aneurysm of the ulnar artery in a child.Injury. 1997; 28: 401-403Abstract Full Text PDF PubMed Scopus (11) Google Scholar; of these, only one was in an infant.3Offer G.J. Sully L. Congenital aneurysm of the ulnar artery in the palm.J Hand Surg (Br). 1999; 24: 735-737Crossref PubMed Scopus (23) Google Scholar Cron et al6Cron J. Saliou C. Fabiani J.N. Traumatic aneurysm of the ulnar artery in a child.Injury. 1997; 28: 401-403Abstract Full Text PDF PubMed Scopus (11) Google Scholar reported a case of a traumatic ulnar artery aneurysm in a child who sustained a closed fracture of the distal radius and ulna. The ulnar artery aneurysm was resected without vascular reconstruction because of adequate intraoperative hand perfusion after resection.6Cron J. Saliou C. Fabiani J.N. Traumatic aneurysm of the ulnar artery in a child.Injury. 1997; 28: 401-403Abstract Full Text PDF PubMed Scopus (11) Google Scholar In two of these reports, there was no history of trauma, and the ulnar artery aneurysm was thought to be congenital in origin.4Deune E.G. McCarthy E.F. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis.Orthopedics. 2005; 28: 1459-1461PubMed Google Scholar, 5Witt P.D. Bowen K.A. Johansen K. True ulnar artery aneurysm of the hand in an 8-year-old boy.Plast Reconstr Surg. 2003; 111: 2475-2476PubMed Google Scholar A reversed saphenous vein interposition graft was used in one patient5Witt P.D. Bowen K.A. Johansen K. True ulnar artery aneurysm of the hand in an 8-year-old boy.Plast Reconstr Surg. 2003; 111: 2475-2476PubMed Google Scholar to reconstruct the ulnar artery, and in the other patient,4Deune E.G. McCarthy E.F. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis.Orthopedics. 2005; 28: 1459-1461PubMed Google Scholar the reconstruction was done with an ipsilateral dorsal hand vein interposition graft. The reconstruction in these two cases was done because the hand was not adequately perfused after the aneurysm resection. Offer and Sully3Offer G.J. Sully L. Congenital aneurysm of the ulnar artery in the palm.J Hand Surg (Br). 1999; 24: 735-737Crossref PubMed Scopus (23) Google Scholar reported a case of an ulnar artery aneurysm in a 1-year-old boy who had no history of trauma. The child had been managed nonoperatively; however, pain and sensory dysfunction developed after 1 year of follow-up. He underwent aneurysm resection without vascular reconstruction because of adequate intraoperative hand perfusion after resection. The cause of the ulnar artery aneurysm in the presented case is not known. The normal physical appearance of the patient, normal values of the inflammatory markers, and the histologic picture of the aneurysm may exclude the possibility that this aneurysm is related to connective tissue disorders or inflammatory conditions. Although the parents reported no trauma, this toddler could have sustained unwitnessed blunt trauma to the hand from falls during the time when he was learning how to walk. The diagnostic and treatment algorithms for ulnar artery aneurysm are not well established because the disease is very rare. Some diagnostics methods have been highlighted in this case. Vascular laboratory tests such as Doppler ultrasound and arterial duplex scanning pose many advantages:•The tests are not invasive and do not require general anesthesia in infants.•They can delineate and localize the ulnar artery aneurysm with accuracy and they can identify the presence of mural thrombus.•They are of great help in the assessment of the adequacy of hand perfusion perioperatively and therefore help in the decision of whether to ligate or to reconstruct the aneurysm. Magnetic resonance imaging (MRI) has been advocated in the diagnostic work-up for palmar artery aneurysms,4Deune E.G. McCarthy E.F. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis.Orthopedics. 2005; 28: 1459-1461PubMed Google Scholar, 5Witt P.D. Bowen K.A. Johansen K. True ulnar artery aneurysm of the hand in an 8-year-old boy.Plast Reconstr Surg. 2003; 111: 2475-2476PubMed Google Scholar but it has some disadvantages. These include the requirement of general anesthesia and the low quality in visualizing digital arteries, as shown by Deune and McCarthy.4Deune E.G. McCarthy E.F. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis.Orthopedics. 2005; 28: 1459-1461PubMed Google Scholar Although conventional arteriogram is an invasive procedure and requires general anesthesia, it can visualize small arteries and aid in the planning of surgery, especially with selective radial and ulnar arteries angiogram. In addition, it can be used as a therapeutic option in the form of embolization of favorable lesions. Recommending MRI or conventional arteriogram in the work-up of an ulnar artery aneurysm in children cannot be drawn from anecdotal case reports, including this report. The choice between these modalities should be based on the availability, presence of expertise, and the possibility of endovascular intervention. Treatment of ulnar artery aneurysms should not be determined by the presence of symptoms. The development of thromboembolic complications with subsequent finger and hand ischemia can occur without warning signs.7Dalman R.L. Upper extremity arterial bypass distal to the wrist.Ann Vasc Surg. 1997; 11: 550-557Abstract Full Text PDF PubMed Scopus (20) Google Scholar The surgical options for ulnar artery aneurysms depend on the presence of adequate perfusion in the hand after the aneurysm is excluded from the hand circulation. Simple resection is the surgical option if the hand is adequately perfused and the radial artery is intact; however, if the hand perfusion is inadequate, ulnar artery reconstruction using microsurgical technique is mandatory.8Rothkopf D.M. Bryan D.J. Cuadros C.L. May Jr, J.W. Surgical management of ulnar artery aneurysms.J Hand Surg (Am). 1990; 15: 891-897Abstract Full Text PDF PubMed Scopus (42) Google Scholar The reconstruction can be achieved by a primary end-to-end anastomosis if there is no tension or with the use of an interposition vein graft if the defect is large. Some authors believe that the ulnar artery should be reconstructed whenever it is resected because this will prevent future complications if the radial artery is compromised by trauma or degenerative arterial disease.4Deune E.G. McCarthy E.F. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis.Orthopedics. 2005; 28: 1459-1461PubMed Google Scholar In the present case, the ulnar artery aneurysm was resected without reconstruction because of the angiographic evidence that the radial artery was the dominant artery of the hand, normal preoperative Allen test, normal preoperative finger pressure measurement with ulnar artery occlusion, and intraoperative evidence of adequate hand perfusion after excluding the aneurysm from the hand circulation as documented by good Doppler signals in all digital arteries.