Title: An unusual case of the syndrome of cervical rib with subclavian artery thrombosis and cerebellar and cerebral infarctions

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1 Author's response to reviews Title: An unusual case of the syndrome of cervical rib with subclavian artery thrombosis and cerebellar and cerebral infarctions Authors: Mirza Jusufovic Else C Sandset (e.c.sandset@medisin.uio.no) Trine H Popperud (tripop@ous-hf.no) Steinar Solberg (ssolber@online.no) Geir Ringstad (gringsta@ous-hf.no) Emilia Kerty (emilia.kerty@medisin.uio.no) Version: 3 Date: 18 April 2012 Author's response to reviews: see over

2 Oslo University Hospital Department of Neurology P.O. Box 4950 Nydalen N-0424 Oslo Norway Switch board: Org. number: NO MVA To Prof Patrícia Canhão, Editor of BMC Neurology Your ref.: Our ref.: Date: Subject: Manuscript # An unusual case of the syndrome of cervical rib with subclavian artery thrombosis and cerebellar and cerebral infarctions Dear Patrícia Canhão, Please thank the reviewers for their helpful comments on our paper. Both reviewers comments have been carefully addressed as described below. We have made substantial changes to the article as suggested by the reviewers. I hereby confirm that all authors have read the manuscript, the paper has not been previously published, and is not under simultaneous consideration by another journal. In addition, all persons involved in the writing of the manuscript have been included on the author list. I hope that the revised manuscript can be accepted for publication in BMC Neurology. Sincerely, Mirza Jusufovic, MD (sign.) Department of Neurology Oslo University Hospital Postal address: Rikshospitalet P.O. Box 4950 Nydalen N-0424 Oslo, Norway Tel: (cell) Fax: mirzajus@hotmail.com

3 A point-by-point response to the concerns Reviewer: Louis Caplan Level of interest: An article whose findings are important to those with closely related research interests. Major Compulsory Revisions 1. The patient did not have the thoracic outlet syndrome. The cervical rib compressed the subclavian artery leading to thrombosis. This has been well reported in the past. I suggest omitting the term thoracic outlet syndrome. We agree with the reviewer that it is of interest to know about cerebrovascular complications due to the syndrome of cervical rib with subclavian artery thrombosis, even though they are unusual. We agree with the reviewer that the term the thoracic outlet syndrome should be understressed. We apologize for the mistake. Title (An unusual case of the syndrome of cervical rib with subclavian artery thrombosis and cerebellar and cerebral infarctions), introduction and parts of discussion are re-written with more emphasis on the syndrome of cervical rib with subclavian arterial thrombosis and cerebral embolism and less emphasis on the thoracic outlet syndrome. 2. The historical review omits the early work on the subject. In addition to your ref 8. it was Charles Symonds who popularized the topic. Symonds CP. Two cases of thrombosis of subclavian artery with contralateral hemiplegia of sudden onset, probably embolic. Brain 1927;50: The article by Charles Symonds was an important early work on the subject. Regrettably, we failed to include it in the manuscript. We have, of course, included this reference in the revision (introduction). Gould AP. A case of spreading obliterative arteritis. Trans Clin Soc London 1884;17:95. Gould AP. Further notes of a case of obliterative arteritis. Trans Clin Soc London 1987;20:252. Hoobler SW. The syndrome of cervical rib with subclavian arterial thrombosis and hemiplegia due to cerebral embolism. N Engl J Med 1942;226: Thanks, we consider these articles important and they are included in the revision (discussion). Keen WW. The symptomatology, diagnosis, and surgical treatment of cervical ribs. Am J Med Sci 1907;133: Halstead WS. An experimental study of circumscribed dilatation of an artery immediately distal to a partially occluding band and its bearing on the dilatation of the subclavian artery observed in certain cases of cervical rib. J Exper Med 1916;24: Thanks, we consider these articles important but consider the early work (see previous answer) as sufficient to highlight main points about the syndrome of cervical rib with subclavian arterial thrombosis and cerebral embolism.

4 Minor Essential Revisions The lesion can involve the Innominate artery or the subclavian. We agree with the reviewer and have included this in the revision. The syndrome of occlusion is more common in athletes and workers, baseball pitchers, golfers, and cricket bowlers in whom arm motions encourage contact with the artery and rib This information is now added. Reviewer: George L. Hines This is an interesting case that describes a phenomenon that has previously been reported. An article of limited interest. The main purpose of the paper has been to describe cerebrovascular complications due to the syndrome of cervical rib with subclavian artery thrombosis. These complications are unusual and are of interest to both neurologists and vascular surgeons in the interdisciplinary process of management of such patients. Although I think it is worthwhile presenting this in a forum viewed by neurologists (as opposed to strictly vascular surgeons) I do believe there are several controversial points in the management of this patient. First I believe that an occlusion of the subclavian artery in a young man should have been more thoroughly evaluated at initial presentation and not just treated with antiplatelets agents. We agree with the reviewer at this point. However, six months prior to recent cerebrovascular complication, the patient was admitted to a local and not our hospital due to a right brachial artery occlusion. Conservative treatment with aspirin 75 mg was initiated than. Unfortunately these procedures were not performed. We have pointed this out in the first part of the discussion. Secondly I think that at the time of rib resection, arterial continuity should have been reestablished both to provide improved perfusion of the arm and to prevent possible further retrograde embolization. In our case, removal of the right cervical rib was deferred in order to prevent collapse of the subclavian thrombus and further retrograde thromboembolization. The patient did not have any vascular sequelae and was initially treated with combined anti-coagulant and anti-platelet therapy and followed closely at regular intervals. We chose, as described in the article, another treatment approach in this case. In general we agree with the reviewer that the subclavian artery should be examined and opened. The supraclavicular approach is necessary for this. If the artery contains thrombus, it is removed during the exploration. Extension into the carotid can also be removed at the same time. If there is carotid thrombus, it is advisable to obtain control of the carotid artery distal to the thrombus to avoid additional embolization to the brain when performing carotid thromboemoblectomy.

5 However, we stress in the conclusion that surgical removal, instead of conservative treatment, should be considered in a patient with subclavian artery compression due to a cervical rib to prevent additional embolic events.

An unusual case of the syndrome of cervical rib with subclavian artery thrombosis and cerebellar and cerebral infarctions

Jusufovic et al. BMC Neurology 2012, 12:48 CASE REPORT Open Access An unusual case of the syndrome of cervical rib with subclavian artery thrombosis and cerebellar and cerebral infarctions Mirza Jusufovic