Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 2  |  Page : 187-189

Idiopathic proximal ulnar artery aneurysm


Jain Institute of Vascular Sciences, A Unit of Bhagwan Mahaveer Jain Hospital, Bengaluru, Karnataka, India

Date of Submission18-Dec-2018
Date of Acceptance18-Mar-2019
Date of Web Publication17-Jun-2020

Correspondence Address:
Dr. S Roshan Rodney
Jain Institute of Vascular Sciences, A Unit of Bhagwan Mahaveer Jain Hospital, Bengaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijves.ijves_95_18

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  Abstract 


Ulnar artery aneurysms are very rare, usually caused by repetitive blunt trauma to the ulnar artery, leading to the formation of an aneurysm. Other etiologies reported in the literature were vasculitis, anatomic abnormalities, or infections. This case report is regarding a 29-year-old male who presented with proximal ulnar artery aneurysm with distal embolization. He underwent aneurysm resection with ligation of the ulnar artery. Histopathology of the aneurysmal wall showed true aneurysm of the ulnar artery.

Keywords: Aneurysms, embolization, ligation, vasculitis


How to cite this article:
Rodney S R, Anand V, Vishnu M, Raj S, Chaudhari H, Sravan C P, Lende V, Vishal H, Krishna K S, Nishan B. Idiopathic proximal ulnar artery aneurysm. Indian J Vasc Endovasc Surg 2020;7:187-9

How to cite this URL:
Rodney S R, Anand V, Vishnu M, Raj S, Chaudhari H, Sravan C P, Lende V, Vishal H, Krishna K S, Nishan B. Idiopathic proximal ulnar artery aneurysm. Indian J Vasc Endovasc Surg [serial online] 2020 [cited 2020 Jul 5];7:187-9. Available from: http://www.indjvascsurg.org/text.asp?2020/7/2/187/286918




  Introduction Top


Ulnar artery aneurysms are rare, most commonly occurring in the distal ulnar artery close to the palmar arch.[1],[2] In 1970, Conn et al. described this form of ulnar aneurysm secondary to repeated palmar trauma as “hypothenar hammer syndrome.”[3] Proximal ulnar artery aneurysms are even less frequently reported. Here, we describe a rare case of proximal ulnar artery aneurysm resulting in acute upper-limb ischemia which was successfully treated surgically.


  Case Report Top


A 29-year-old patient, dermatologist by profession, nonsmoker with no comorbidities presented to our outpatient department with complaints of gradually increasing right hand and forearm pain of 4-day duration. There was no history of trauma or iatrogenic injury to his right upper limb. He had no history of intravenous drug use. On examination, he was noted to have a lumpiness in his ulnar side of the proximal forearm, nontender and nonpulsatile with palpable radial and nonpalpable ulnar arteries. The Allen's test was positive with no other ischemic signs in the right upper limb. Duplex ultrasound was done which demonstrated a proximal ulnar artery aneurysm of size 12 mm × 5 mm with significant intra-aneurysmal thrombus load with nonocclusive thrombus in the distal ulnar artery but with patent palmar arch [Figure 1]. Computed tomography (CT) angiogram of the arch of the aorta with the right upper limb was done which showed aneurysmal focal dilatation of the ulnar artery in the proximal forearm with intraluminal thrombus
Figure 1: Ultrasound image (a) and ultrasound Doppler image (b) demonstrating ulnar artery aneurysm, patent, but with significant intra-aneurysmal thrombus

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[Figure 2]. Cervical spine X-ray showed no evidence of cervical rib. Two-dimensional echo was found to be normal. A diagnosis of ulnar artery aneurysm with distal embolization was made. Systemic anticoagulation was started in the form of therapeutic low-molecular-weight heparin administered subcutaneously, and the patient was planned for definitive operative repair. The ulnar artery aneurysm was approached via a curvilinear incision in the right forearm. Aneurysm was gently dissected from the surrounding structures [Figure 3] and excised with distal ulnar thrombectomy. The caliber of the ulnar artery was too small for reconstruction. Since the preoperative CT angiogram showed normal radial artery with patent palmar arch, proximal and distal ulnar artery ligation was done without an attempt for reconstruction. After the resection and distal thrombectomy, both the ulnar and radial pulses were palpable with strong Doppler signals. The surgery was uneventful, and he recovered well with no signs of upper-limb ischemia or loss of function. The excised aneurysm tissue and thrombus were sent for culture and histopathological examination [Figure 4]. Culture was negative, and histopathology revealed a true aneurysm of the ulnar artery. Further imaging for other aneurysms including aortoiliac and femoropopliteal was negative. Rheumatology evaluation to rule out vasculitic and connective tissue disorders was found to be negative.
Figure 2: Computed tomography angiogram demonstrating (a) aneurysm of the ulnar artery (small arrows) with (b) three-dimensional reconstruction

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Figure 3: Ulnar artery aneurysm identified and isolated

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Figure 4: Resected aneurysm sent for histopathology examination

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  Discussion Top


Ulnar artery aneurysms are rare, usually found in young males, and often occur with symptoms and signs of distal ischemia. Hippocrates in 460 BC was the first to diagnose an upper-extremity arterial aneurysm. The first description of an ulnar artery aneurysm in the palm was by Guattani et al. in 1772.[4]

Upper-extremity arterial aneurysms are uncommon lesions, and are most commonly false aneurysms. The natural history of ulnar artery aneurysms is not well established due to the small number of cases reported. They arise from repetitive blunt trauma, vasculitis, vascular anomalies, or infections of the upper extremity. Patients may present with ischemic symptoms or numbness and tingling along the fourth and fifth digits of the right hand due to compression of the ulnar nerve.[5] The severity of symptoms is based on the location of the obstruction and the nature of collateral hand circulation. Spontaneous formation occurs with acute hypertension and sudden change to blood flow causing intimal tears. Distal ulnar artery aneurysms, although uncommon, have been described in adults as a part of the hypothenar hammer syndrome. Repetitive blunt trauma damages the intima of the ulnar artery as it passes adjacent to the hook of the hamate. Subsequently, the artery becomes aneurysmal or thrombose, affecting digital arteries. Ulnar artery aneurysms can be either true or false, depending on the presence or absence of attenuated layers of the normal arterial wall in the aneurysm. False aneurysms in the upper extremity are most commonly due to sharp penetrating trauma.[6] True ulnar artery aneurysms involving the more proximal ulnar artery have been previously reported associated with vasculitic disorders. These have included true ulnar aneurysms in the setting of Behcet's disease,[7] rheumatic vasculitis,[8] and eosinophilia.[9] There has also been a reported association with Marfan's syndrome, where Nguyen described hypothenar ulnar aneurysm in a 50-year-old male accountant.[10] Mycotic aneurysms of the ulnar artery have also been reported, with the first case reported in 1987 secondary to Streptococcus bovis septicemia.[11]

The diagnostic workup of ulnar artery aneurysms includes a duplex ultrasound scan, conventional angiography, multidetector CT angiography, and magnetic resonance angiography.[12] Upper-extremity arterial aneurysms have long been managed by operative intervention since the first report by Griffith in 1897. Surgical options for ulnar artery aneurysms include arterial ligation (assuming an intact palmar/radial arch), resection of the thrombosed arterial segment or aneurysm with end-to-end anastomosis, and resection and vascular reconstruction with an autogenous vein or artery graft.[13]

The need for revascularization after resection of the aneurysm is controversial. Selective revascularization is sometimes advocated based on certain indications. These include the lack of adequate hand perfusion, the location of the aneurysm, presence of favorable superficial palmar arch anatomy for reconstruction, and the status of collateral hand circulation.[14]

Simple resection is the surgical option if the forearm/hand is adequately perfused and the radial artery is intact; however, if the forearm/hand perfusion is inadequate, ulnar artery reconstruction is mandatory.[13]

This patient most likely had a true spontaneous aneurysm as the patient's history did not fit the criteria for any infectious cause, and no systemic disabling symptoms, including joint pain, were reported. The histopathological examination confirmed it as a true aneurysm with intimal disruption with medial degeneration and myxoid change.


  Conclusion Top


Ulnar artery aneurysms are extremely rare, and occur most commonly in the palm, often secondary to repetitive trauma. Diagnosis of a proximal ulnar artery aneurysm may represent a diagnostic challenge given its rarity. The management is, however, similar with repair recommended given the risk of embolic complications.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
McHugh SM, Moloney MA, Greco E, Wheatcroft M. True ulnar artery aneurysm in the proximal forearm: Case report and literature review. Ann Vasc Surg 2017;44:421.e1-4.  Back to cited text no. 1
    
2.
Conn J Jr., Bergan JJ, Bell JL. Hypothenar hammer syndrome: Posttraumatic digital ischemia. Surgery 1970;68:1122-8.  Back to cited text no. 2
    
3.
Mazzaccaro D, Malacrida G, Stegher S, Occhiuto MT, Caldana M, Tealdi DG, et al. Ulnar artery aneurysm: Case report and review of the literature. G Chir 2012;33:110-3.  Back to cited text no. 3
    
4.
Gray RJ, Stone WM, Fowl RJ, Cherry KJ, Bower TC. Management of true aneurysms distal to the axillary artery. J Vasc Surg 1998;28:606-10.  Back to cited text no. 4
    
5.
Nickul N, Shah MS 4th, David Roman RP, Roland Purcell MD. Aberrant ulnar artery and ulnar artery thrombosis with nerve entrapment: A case report and review of literature. J Vasc Med Surg 2015;3:230.  Back to cited text no. 5
    
6.
De Santis F, Martini G, Mani G, Zywica M, Zipponi D. Forearm and hand arteries' aneurysms – A case report of bilateral true ulnar artery aneurysm in the hypothenar eminence and systematic review of the literature. Vascular 2013;21:169-76.  Back to cited text no. 6
    
7.
Kisacik B, Kasifoglu T, Akay S, Yilmaz O, Yilmaz S, Simsek I, et al. Ulnar artery aneurysm in a patient with Behçet's disease. Rheumatol Int 2010;30:383-5.  Back to cited text no. 7
    
8.
Makarenko TP, Klimkovich IG. Spontaneous aneurysm of the ulnar artery as a complication of rheumatic vasculitis. Khirurgiia (Mosk) 1958;34:109-11.  Back to cited text no. 8
    
9.
Sekino S, Takagi H, Kato T, Matsuno Y, Sekido Y, Umemoto T, et al. Nontraumatic pseudoaneurysm of the proximal ulnar artery with eosinophilia. J Vasc Surg 2005;42:1233-5.  Back to cited text no. 9
    
10.
Nguyen DQ, Murison M. Ulnar artery aneurysm in a patient with Marfan's syndrome. J Plast Reconstr Aesthet Surg 2006;59:1131-2.  Back to cited text no. 10
    
11.
Bacourt F, Bornet P, Fourcade R, Davous N, Lacombe P. Aneurysm of the ulnar artery in Streptococcus bovis septicemia. First case. Presse Med 1987;16:450.  Back to cited text no. 11
    
12.
Clagett PG. Upper extremity aneurysms. In: Rutherford RB, editor. Vascular Surgery. 6th ed. Philadelphia: Elsevier Saunders; 2005. p. 1551-65.  Back to cited text no. 12
    
13.
Rajakumar AP, Saravanan S, Kalidoss L, Rajan S. Proximal ulnar artery aneurysm following a Bentall procedure for type A aortic dissection. J Card Surg 2016;31:742-4.  Back to cited text no. 13
    
14.
Cotroneo A, Barillà D, Armienti F, Martinelli GL, Diena M. Distal ulnar artery aneurysm repair by end-to-end technique: A case report. Ann Vasc Surg 2018;46:367.e15-9.  Back to cited text no. 14
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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