Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 6  |  Issue : 4  |  Page : 324-326

A case report of ulnar artery aneurysm in a 5-month-old infant with a review of literature


1 Department of Vascular and Endovascular Surgery, Ruby Hall Clinic, Pune, Maharashtra, India
2 Department of Paediatric Surgery, Ruby Hall Clinic, Pune, Maharashtra, India
3 Department of Paediatrics, Ruby Hall Clinic, Pune, Maharashtra, India

Date of Submission12-Apr-2019
Date of Acceptance13-May-2019
Date of Web Publication20-Dec-2019

Correspondence Address:
Bhushan D Shinde
Department of Vascular and Endovascular Surgery, Ruby Hall Clinic, Pune, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijves.ijves_23_19

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  Abstract 


Aneurysms in the upper extremity vascular system are rare and exceedingly uncommon in young infants. We present a case report of a 5-month-old infant with multiple aneurysms in proximal left ulnar artery. Trauma as etiology was ruled out based on history. Detailed investigation did not reveal any particular etiology. Surgical management involved excision of aneurysm and graft placement. Excision biopsy confirmed giant-cell arteritis on histopathology.

Keywords: Aneurysm, giant cell arteritis, ulnar artery


How to cite this article:
Kamerkar DR, Ratta B S, Datla S, Kuntilla N, Shinde BD, Sawant V. A case report of ulnar artery aneurysm in a 5-month-old infant with a review of literature. Indian J Vasc Endovasc Surg 2019;6:324-6

How to cite this URL:
Kamerkar DR, Ratta B S, Datla S, Kuntilla N, Shinde BD, Sawant V. A case report of ulnar artery aneurysm in a 5-month-old infant with a review of literature. Indian J Vasc Endovasc Surg [serial online] 2019 [cited 2020 Feb 27];6:324-6. Available from: http://www.indjvascsurg.org/text.asp?2019/6/4/324/273590




  Introduction Top


Ulnar artery aneurysms in general population are very rare. (5) Ulnar artery aneurysm may develop for traumatic, atherosclerotic and infectious reasons. Distalulnar artery aneurysm although uncommon have been well described in adults as Hypothenar Hammer syndrome (5).


  Case Report Top


A 5-month-old infant presented to us with a pulsatile mass over the left upper forearm. There were no signs or symptoms suggestive of thromboembolic events. Birth history and previous medical history were unremarkable. There was no history suggestive of any trauma or penetrating injury to the upper limb [Figure 1].
Figure 1: Computed tomography arteriogram of the left upper limb

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On physical examination, the infant was in a healthy condition with no dysmorphism. The left upper limb examination showed a 1.5-cm pulsatile, nontender, compressible mass in the inner aspect of the left forearm. The radial and ulnar arteries were palpable at the wrist with a normal Allen test. There were no signs of ischemia.

Routine laboratory investigations, including complete blood count, C-reactive protein, and erythrocyte sedimentation rate, done were within normal limits. Immunological markers such as antinuclear antibody, antineutrophil cytoplasmic antibody, and perinuclear antineutrophil cytoplasmic antibody were negative.

An arterial duplex study revealed a saccular aneurysm measuring 1.3 cm × 1.1 cm × 1.0 cm seen from the proximal ulnar artery in the forearm with an eccentric thrombus within.

Contrast tomography arteriogram was suggestive of multiple variable-sized aneurysms of the proximal left ulnar artery located adjacent to each other. The largest aneurysm was measured 1.2 cm × 0.9 cm. The affected ulnar artery segment measured 3 cm in length.

The rest of the ulnar artery was normal [Figure 2].
Figure 2: Computed tomography arteriogram 2ndimage

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Computed tomography arteriogram showed an aneurysm of the ulnar artery and no evidence of aneurysms in the rest of the body [Figure 3].
Figure 3: Clinical photograph showing ulnar artery aneurysm left upper limb

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During surgical exploration, a longitudinal incision was made on the area parallel to the area of the aneurismal artery. Dissection through the fascial layers revealed a 1.5-cm ulnar artery aneurysm.

A trial of intraoperative clamping of the aneurysm proximally and distally resulted in 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 sacs were resected.

After identification of the ulnar nerve, the dissection was done, and the vessel loops were used to control the aneurysm proximally and distally [Figure 4]. The aneurysmal segment was resected and replaced with vein (long saphenous vein from the thigh) interposition graft placement with intermittent sutures, the decision was taken to repair ulnar artery with interposition vein graft, as very short segment of the ulnar artery was aneurysmal with a distal and proximal segment of artery disease free.
Figure 4: Intraoperative photo showing short segment aneurysmal dilatation of ulnar artery

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Postoperatively, the patient was started on antiplatelet (tablet ecosprin 12.5 mg) once a day and continued for 3 months.

[Figure 4] shows the ulnar artery aneurysm which was controlled proximally and distally using vessel loops intraoperatively.

Histopathology 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 the elastin fibers in the media. It showed granulomatous inflammation, presence of giant cells, especially in the media, atrophy of the smooth muscles, and destruction of elastic fibers, all features suggestive of giant-cell arteritis.

The child's postoperative course was uneventful and was discharged on the 3rd postoperative day. At follow-up, the patient's left upper limb function and perfusion were normal as evidenced by arterial duplex studies.

The child has been followed up with Doppler evaluation at 1 year of age, and in the study, the venous graft is visualized and shows partially arterialized flow with a velocity of 30–40 cm/s same as proximal and distal segment of the ulnar artery. No thrombosis or pseudoaneurysm was noted. The distal ulnar artery shows normal color filling, triphasic waveform.


  Discussion Top


Only a few cases of childhood ulnar artery aneurysms have been reported till date, in that early infantile age is an extremely rare occurrence.[1],[2],[3],[4]

Ulnar artery aneurysms in the general population are very rare.[5] Ulnar artery aneurysm may develop for traumatic, atherosclerotic, and infectious reasons. Distal ulnar artery aneurysms, although uncommon, have been well described in adults as hypothenar hammer syndrome.[5]

A computerized literature search was conducted in MEDLINE (1966–September 2006) and EMBASE (1980–September 2006). Four cases of ulnar artery aneurysm have been reported in the pediatric population; only one was in infant.[1] Computerized research done by us in PubMed and Medscape from 2007 to 2017 showed five cases in the pediatric population, of which four are nontraumatic etiology.

Several possible etiologies have been reported, including connective tissue and autoimmune disorders such as Ehlers–Danlos syndrome, Marfan syndrome, and Kawasaki disease.[6],[7]

The diagnosis and treatment of ulnar artery aneurysm are not well established, because the disease is very rare. High-resolution duplex color Doppler examination is useful in confirming the diagnosis of the palmar aneurysm. Duplex color Doppler is a noninvasive test, which allows the examiner to evaluate the damage to vessel wall and presence of hematoma. However, angiography is currently the “gold standard” in aneurysmal diagnosis, providing crucial information for preoperative planning such as location, size, and shape of the arterial lesion in question, as well as the presence of any collateral circulation.[8]

Several modalities of treatment for hand aneurysm exist and include both conservative and surgical options. Conservative methods of treatment which include a low-calorie diet, and intravenous administration of prostaglandin E1 and vasodilation agents, have been suggested by some as the first line of therapy.[9]

The surgical options of the ulnar artery aneurysm depend on the presence of adequate perfusion in the hand after the aneurysm is excluded from the 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.[10]

The reconstruction can be achieved by the primary end-to-end anastomosis if there is no tension or with the use of an interposition vein graft if the defect is large.

Deune and McCarthy suggest ulnar artery to be reconstructed always, whenever it is resected to avoid future complications in case of trauma or degenerative disease to ipsilateral radial artery.[2]

The cause of the aneurysm in this child was due to giant-cell arteritis as confirmed by the histopathology report. Giant-cell arteritis also known as temporal arteritis is a systemic vascular syndrome predominantly affecting the cranial arteries. Vascular insufficiency related to stenosis and thrombosis of affected vessels may lead to renovascular hypertension, neurologic symptoms, or extremity claudications. In conclusion, giant-cell arteritis can present as ulnar artery aneurysm though rare.

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.
Offer GJ, Sully L. Congenital aneurysm of the ulnar artery in the palm. J Hand Surg Br 1999;24:735-7.  Back to cited text no. 1
    
2.
Deune EG, McCarthy EF. Reconstruction of a true ulnar artery aneurysm in a 4-year-old patient with radial artery agenesis. Orthopedics 2005;28:1459-61.  Back to cited text no. 2
    
3.
Witt PD, Bowen KA, Johansen K. True ulnar artery aneurysm of the hand in an 8-year-old boy. Plast Reconstr Surg 2003;111:2475-6.  Back to cited text no. 3
    
4.
Cron J, Saliou C, Fabiani JN. Traumatic aneurysm of the ulnar artery in a child. Injury 1997;28:401-3.  Back to cited text no. 4
    
5.
Ferris BL, Taylor LM Jr., Oyama K, McLafferty RB, Edwards JM, Moneta GL. Hypothenar hammer syndrome: Proposed etiology. J Vasc Surg 2000;31:104-13.  Back to cited text no. 5
    
6.
Iyer RS, Hanel DP, Enriquez BK, Weinberger E. Ulnar artery aneurysm causing palmar mass in 5-month-old girl. Pediatr Radiol 2012;42:1401-4.  Back to cited text no. 6
    
7.
Sarkar R, Coran AG, Cilley RE, Lindenauer SM, Stanley JC. Arterial aneurysms in children: Clinicopathologic classification. J Vasc Surg 1991;13:47-56.  Back to cited text no. 7
    
8.
Blum AG, Zabel JP, Kohlmann R, Batch T, Barbara K, Zhu X, et al. Pathologic conditions of the hypothenar eminence: Evaluation with multidetector CT and MR imaging. Radiographics 2006;26:1021-44.  Back to cited text no. 8
    
9.
Mehlhoff TL, Wood MB. Ulnar artery thrombosis and the role of interposition vein grafting: Patency with microsurgical technique. J Hand Surg Am 1991;16:274-8.  Back to cited text no. 9
    
10.
Rothkopf DM, Bryan DJ, Cuadros CL, May JW Jr. Surgical management of ulnar artery aneurysms. J Hand Surg Am 1990;15:891-7.  Back to cited text no. 10
    


    Figures

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



 

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