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SHORT REPORT Endovascular Management of an Unusual Cause of Pancoast Syndrome A.D. Lee, 1 * G.K. Kota, 2 N. Shyamkumar, 2 D.T. Abraham 1 and S. Agarwal 1 Departments of 1 Surgery, and 2 Radiology, Christian Medical College and Hospital, Vellore, India Vascular conditions presenting with Pancoast syndrome are rare. A case of vertebral artery pseudoaneurysm presenting with Pancoast syndrome is reported. The aneurysm was successfully treated by proximal coil embolization. Keywords: Pancoast’s syndrome; Vertebral artery; Aneurysm, false. Pancoast syndrome is a distinct group of signs and symptoms that include shoulder and arm pain, Horner’s syndrome and wasting of muscles of the hand. Local extension of bronchogenic carcinoma of the lung apex (Pancoast tumour) is the commonest cause. 1 We report the case of a 59-year-old man presenting with features of Pancoast syndrome of short duration due to a vertebral artery pseudoaneurysm. The diagnostic difficulty and the endovascular management of the case are discussed. Case Report A 59-year-old man presented with a 1-month history of progressive weakness of his right upper limb with shooting pain from the neck along the arm, hoarseness of voice and swelling in the right supraclavicular region. On examination, he was found to have an 8 by 6 cm tender, hard, non-pulsatile swelling with diffuse margins in the right supraclavicular region. He had no power in the deltoid, triceps, biceps, forearm muscles and intrinsic and extrinsic muscles of the hand. There was complete anesthesia in C5 to T1 dermatomes. He did not have a Horner’s syndrome. The possibility of a Pancoast tumour was considered and a contrast enhanced CT scan of the chest was performed which showed a mass lesion in the right supraclavicular region. An ultrasound-guided biopsy of the mass was carried out and showed chronic inflam- mation and fibrosis. Subsequently, the CT scan was reviewed and the possibility of a pseudoaneurysm of the right vertebral artery was considered (Fig. 1). A Doppler study revealed turbulent flow within the mass lesion and an angiogram demonstrated a fusiform pseudoa- neurysm from the right vertebral artery about 3 cm from its origin. There was no forward flow beyond the pseudoaneurysm into the distal right vertebral artery. There was retrograde filling of the distal right vertebral artery on left vertebral injection, but the pseudoaneur- ysm did not fill. Selective embolisation of the right vertebral artery proximal to the pseudoaneurysm was performed using three pushable coils. Post-embolisation completion angiogram showed total occlusion of the proximal right vertebral artery (Fig. 2(a) and (b)). The patient received physiotherapy for the upper limb weakness. At 6-month follow up, the neck swelling had disappeared and there was significant improve- ment in muscle power of the deltoid, biceps and finger flexors. Repeat duplex of the neck showed a small hypoechoic area corresponding to the site of the aneurysm with no residual flow. Discussion There have been only two reports of vascular conditions presenting with Pancoast syndrome, i.e. a Eur J Vasc Endovasc Surg 30, 621–623 (2005) doi:10.1016/j.ejvs.2005.06.020, available online at http://www.sciencedirect.com on *Corresponding author. Dr A.D. Lee, MS, DNB, Department of General and Vascular Surgery Unit 2, Christian Medical College and Hospital, Vellore 632004, India. E-mail address: [email protected] 1078–5884/000621 + 03 $35.00/0 q 2005 Elsevier Ltd. All rights reserved.

Endovascular Management of an Unusual Cause of Pancoast Syndrome

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SHORT REPORT

*CorrespondiGeneral and VHospital, VelE-mail address

1078–5884/00

Endovascular Management of an Unusual Cause ofPancoast Syndrome

A.D. Lee,1* G.K. Kota,2 N. Shyamkumar,2 D.T. Abraham1 and S. Agarwal1

Departments of 1Surgery, and 2Radiology, Christian Medical College and Hospital, Vellore, India

Vascular conditions presenting with Pancoast syndrome are rare. A case of vertebral artery pseudoaneurysm presentingwith Pancoast syndrome is reported. The aneurysm was successfully treated by proximal coil embolization.

Keywords: Pancoast’s syndrome; Vertebral artery; Aneurysm, false.

Pancoast syndrome is a distinct group of signs andsymptoms that include shoulder and arm pain, Horner’ssyndrome and wasting of muscles of the hand. Localextension of bronchogenic carcinoma of the lung apex(Pancoast tumour) is the commonest cause.1 We reportthe case of a 59-year-old man presenting with features ofPancoast syndrome of short duration due to a vertebralartery pseudoaneurysm. The diagnostic difficulty andthe endovascular management of the case are discussed.

Case Report

A 59-year-old man presented with a 1-month historyof progressive weakness of his right upper limb withshooting pain from the neck along the arm, hoarsenessof voice and swelling in the right supraclavicularregion. On examination, he was found to have an 8 by6 cm tender, hard, non-pulsatile swelling with diffusemargins in the right supraclavicular region. He had nopower in the deltoid, triceps, biceps, forearm musclesand intrinsic and extrinsic muscles of the hand. Therewas complete anesthesia in C5 to T1 dermatomes. Hedid not have a Horner’s syndrome.

The possibility of a Pancoast tumour was consideredand a contrast enhanced CT scan of the chest wasperformed which showed a mass lesion in the right

ng author. Dr A.D. Lee, MS, DNB, Department ofascular Surgery Unit 2, Christian Medical College and

lore 632004, India.: [email protected]

0621 + 03 $35.00/0 q 2005 Elsevier Ltd. All rights reser

supraclavicular region. An ultrasound-guided biopsy ofthe mass was carried out and showed chronic inflam-mation and fibrosis. Subsequently, the CT scan wasreviewed and the possibility of a pseudoaneurysm of theright vertebral artery was considered (Fig. 1). A Dopplerstudy revealed turbulent flow within the mass lesionand an angiogram demonstrated a fusiform pseudoa-neurysm from the right vertebral artery about 3 cm fromits origin. There was no forward flow beyond thepseudoaneurysm into the distal right vertebral artery.There was retrograde filling of the distal right vertebralartery on left vertebral injection, but the pseudoaneur-ysm did not fill. Selective embolisation of the rightvertebral artery proximal to the pseudoaneurysm wasperformed using three pushable coils. Post-embolisationcompletion angiogram showed total occlusion of theproximal right vertebral artery (Fig. 2(a) and (b)). Thepatient received physiotherapy for the upper limbweakness. At 6-month follow up, the neck swellinghad disappeared and there was significant improve-ment in muscle power of the deltoid, biceps and fingerflexors. Repeat duplex of the neck showed a smallhypoechoic area corresponding to the site of theaneurysm with no residual flow.

Discussion

There have been only two reports of vascularconditions presenting with Pancoast syndrome, i.e. a

Eur J Vasc Endovasc Surg 30, 621–623 (2005)

doi:10.1016/j.ejvs.2005.06.020, available online at http://www.sciencedirect.com on

ved.

Fig. 1. Contrast enhanced CT scan of the neck showing rightvertebral artery pseudoaneurysm with marked fibrosis andthrombosis.

Fig. 2. (A) Digital subtraction angiogram showing fusiformaneurysm of the right vertebral artery with no distal flow. (B)Post-coil embolisation angiogram showing successful exclu-sion of the aneurysm.

A. D. Lee et al.622

carotid artery pseudoaneurysm2 and a subclavianartery mycotic aneurysm.3 In the case presentedabove, diagnosis of a pseudoaneurysm could not beestablished during the first consultation due to thenear total thrombosis of the aneurysm. Fortunately,biopsy was performed without any untoward compli-cations. Such an extreme degree of fibrosis andthrombosis is a known feature of pseudoaneurysms.The diagnosis was confirmed by digital subtractionangiography. The angiogram showed no flow beyondthe pseudoaneurysm and no retrograde filling of thepseudoaneurysm. Hence, proximal coil embolisationwas sufficient to cause complete thrombosis, reductionin size and relief from the mass effect. Operativetreatment in this case would have been difficult inview of the size of the aneurysm and risk of injury toimportant structures such as the brachial plexus.

The extracranial vertebral artery is a rare site ofaneurysm development. Spontaneous dissection of thevertebral artery is not uncommon4 and traumaticpseudoaneurysms including those following iatro-genic trauma have been reported.5 In the case wepresent, the etiology of the pseudoaneurysm is notclear. There was no history of previous trauma orevidence of a connective tissue disorder. There was nofever or elevated inflammatory markers to suggest amycotic aneurysm. We speculate that the pseudoa-neurysm occurred following unrecognised trauma to adegenerated, atherosclerotic artery.

Successful endovascular exclusion of vertebralartery aneurysms, as presented here, has beenreported by several authors without any seriouscomplications.6 While the natural history of vertebralartery dissection is better known, there has been onlyone report of spontaneous resolution of a vertebralartery pseudoaneurysm.7 It is conceivable that even inthe case described above, the pseudoaneurysm might

Eur J Vasc Endovasc Surg Vol 30, 12 2005

have thrombosed itself over time. However, in view ofthe nerve compression and features of Pancoastsyndrome, the aneurysm warranted treatment. Directthrombin injection is a useful therapeutic option in thetreatment of pseudoaneurysm8,9 and could potentially

Unusual Cause of Pancoast Syndrome 623

have been used in this case. The concerns of arterialthrombosis and distal embolization with the pro-cedure would not have applied to this case, as therewas no flow in the distal artery. Our lack of experienceof thrombin injection precluded its use in our case.

In conclusion, this case highlights an unusual causeof Pancoast syndrome, which was successfully man-aged by endovascular means. A thrombosed pseu-doaneurysm can present as a space-occupying lesionand lead to misdiagnosis.

References

1 Tsao JW, Garlin AB, Marder SR. Pancoast’s syndrome. N EnglJ Med 1998;338(11):765–766.

2 Rong SH. Carotid pseudoaneurysm simulating Pancoast tumor.AJR Am J Roentgenol 1984;142:495–496.

3 Tsao JW, Garlin AB, Marder SR, Haber RJ. Mycotic aneurysmpresenting as Pancoast’s syndrome in an injection drug user. AnnEmerg Med 1999;34:546–549.

4 Chiche L, Praquin B, Koskas F, Kieffer E. Spontaneousdissection of the extracranial vertebral artery: Indications andlong-term outcome of surgical treatment. Ann Vasc Surg 2005;19(1):5–10.

5 Cihangiroglu M, Rahman A, Yildirim H, Burma O, Uysal H.Iatrogenic vertebral artery pseudoaneurysm: US, CT and MRIfindings. Eur J Radiol 2002;43(1):14–18.

6 Barr JD, Lemley TJ. Endovascular arterial occlusion accom-plished using microcoils deployed with and without proximalflow arrest: Results in 19 patients. AJNR Am J Neuroradiol 1999;20(8):1452–1456.

7 Hiatt JR, Martin NA, Machleder HI. The natural history of atraumatic vertebral artery aneurysm: Case report. J Trauma 1989;29(11):1592–1594.

8 Lonn L, Olmarker A, Geterud K, Risberg B. Prospectiverandomized study comparing ultrasound-guided thrombin injec-tion to compression in the treatment of femoral pseudoaneurysms.J Endovasc Ther 2004;11(5):570–576.

9 Jeganathan R, Harkin DW, Lowry P, Lee B. Iatrogenicsubclavian artery pseudoaneurysm causing airway compromise:Treatment with percutaneous thrombin injection. J Vasc Surg 2004;40(2):371–374.

Accepted 10 June 2005Available online 10 August 2005

Eur J Vasc Endovasc Surg Vol 30, 12 2005