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      Multivessel Thromboembolism Associated with Dysfunction of Protein S

      case-report

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          Abstract

          Protein S is a vitamin K-dependent coagulation factor that acts as an anticoagulant. Deficiency of protein S increases the risk of thromboembolic events. We report a case of isolated protein S deficiency in a 39-year-old woman suffering arterial occlusion in both lower legs. She underwent a surgical procedure using thrombectomy and balloon angioplasty of her left lower extremity. Later, she had right trans-tibial amputation because of the reperfusion injury. Throughout the evaluation of thromboembolic events, we diagnosed a large thrombus in the right atrium and an asymptomatic pulmonary thromboembolism. The patient was successfully treated with right atrial thrombectomy and systemic anticoagulation. Careful evaluation for protein S levels may be necessary in patients with arterial thromboembolic events, especially young adults.

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          Most cited references12

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          The European Cooperative Study on the clinical significance of right heart thrombi. European Working Group on Echocardiography.

          G Kronik, G Kronick (1989)
          Information on a total of 119 patients with echocardiographically detected right heart thrombi was collected by questionnaire. Two major thrombus types with different morphology, etiology and clinical significance were identified: (1) 48 patients had long, thin, extremely mobile thrombi which resembled a worm or a snake (type A); (2) 57 patients had more or less immobile, non-specific clots resembling left heart thrombi (type B). Type A patients had a high incidence of deep venous thrombosis and a low incidence of potentially thrombogenetic cardiac abnormalities. The reverse was true for group B. These observations and the peculiar worm-shape of type A thrombi suggest that most type A thrombi originate in peripheral veins, while most type B thrombi develop within the right heart chambers. Clinically, type A patients were a high-risk group; pulmonary embolism was the rule and was usually severe. Early (less than or equal to 8 days) thrombus-related mortality was excessively high (42%), including 13 deaths from pulmonary embolism, one from paradoxical peripheral embolism and six perioperative deaths. Type B thrombi were much more benign; pulmonary embolism was not uncommon (40%) but never fatal. Early thrombus-related mortality was only 4% (two peri-operative deaths). Fourteen cases could not be classified as A or B because their thrombi were highly mobile (= not B) but not worm-shaped (= not A). This small group was intermediate between groups A and B in all respects. An analysis of the relationship between therapy and outcome revealed that type B thrombi had a good prognosis irrespective of the treatment. In type A cases early thrombus-related mortality was much lower with surgery (27%) than with conservative treatment (54%). However, the selection of surgical cases was certainly very biased and it is not clear to what extent the better results of surgery are caused by patient selection. Thus the optimal management of these cases remains to be determined.
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            A study of Protein S antigen levels in 3788 healthy volunteers: influence of age, sex and hormone use, and estimate for prevalence of deficiency state.

            Gary A Dykes, Anita Walker, Katherine McMahon … (2001)
            Total Protein S (tPS) and free Protein S (fPS) antigen levels were measured in 3788 healthy blood donors. Men had higher levels of both parameters than women (P < 0.001). Age had no effect on tPS in men, although there was a slight reduction in fPS levels with increasing age. In women increasing age was associated with a significant increase in tPS levels (P < 0.001) but had no effect on fPS after adjustment for menopausal state. Oral contraceptive pill (OCP) use significantly lowered tPS but had no effect on fPS. In post-menopausal women, hormone replacement therapy (HRT) use had no statistically significant effect on either tPS or fPS. Donors with tPS or fPS levels in the lowest percentile (n = 56) were retested; only nine with repeat low levels were identified, eight of whom had persistently low levels over a 4-7-year follow-up. Acquired deficiency was excluded. When possible, family studies were performed, leading to an estimate of prevalence of familial PS deficiency of between 0.03% and 0.13% in the general population.
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              The frequency of type I heterozygous protein S and protein C deficiency in 141 unrelated young patients with venous thrombosis.

              Matt Griffin, I Scharrer, V Hach-Wunderle … (1988)
              The frequency of heterozygous protein C and protein S deficiency, detected by measuring total plasma antigen, in a group (n = 141) of young unrelated patients (less than 45 years old) with venous thrombotic disease was studied and compared to that of antithrombin III, fibrinogen, and plasminogen deficiencies. Among 91 patients not receiving oral anticoagulants, six had low protein S antigen levels and one had a low protein C antigen level. Among 50 patients receiving oral anticoagulant therapy, abnormally low ratios of protein S or C to other vitamin K-dependent factors were presented by one patient for protein S and five for protein C. Thus, heterozygous Type I protein S deficiency appeared in seven of 141 patients (5%) and heterozygous Type I protein C deficiency in six of 141 patients (4%). Eleven of thirteen deficient patients had recurrent venous thrombosis. In this group of 141 patients, 1% had an identifiable fibrinogen abnormality, 2% a plasminogen abnormality, and 3% an antithrombin III deficiency. Thus, among the known plasma protein deficiencies associated with venous thrombosis, protein S and protein C deficiencies (9%) emerge as the leading identifiable associated abnormalities.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Ann Rehabil Med
                Journal ID (iso-abbrev): Ann Rehabil Med
                Journal ID (publisher-id): ARM
                Title: Annals of Rehabilitation Medicine
                Publisher: Korean Academy of Rehabilitation Medicine
                ISSN (Print): 2234-0645
                ISSN (Electronic): 2234-0653
                Publication date (Print): June 2012
                Publication date (Electronic): 30 June 2012
                Volume: 36
                Issue: 3
                Pages: 414-417
                Affiliations
                Department of Rehabilitation Medicine, College of Medicine, The Catholic University of Korea, Suwon 442-723, Korea.
                [1 ]Department of Surgery, College of Medicine, The Catholic University of Korea, Suwon 442-723, Korea.
                Author notes
                Corresponding author: Seong Hoon Lim. Department of Physical Medicine and Rehabi litation, St. Vincent Hospital, College of Medicine, The Catholic University of Korea, 93-6, Jidong, Suwon 442-723, Korea. Tel: +82-31-249-7650, Fax: +82-31-251-4481, seonghoon@ 123456catholic.ac.kr
                Article
                DOI: 10.5535/arm.2012.36.3.414
                PMC ID: 3400884
                PubMed ID: 22837980
                SO-VID: 4bb84243-ad7c-48b0-b42f-03d91afe38c4
                Copyright © Copyright © 2012 by Korean Academy of Rehabilitation Medicine
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 18 February 2011
                Date accepted : 12 July 2011
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Medicine
                Keywords: protein s,thromboembolism,trans-tibial amputation
                Data availability:
                ScienceOpen disciplines: Medicine
                Keywords: protein s, thromboembolism, trans-tibial amputation

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