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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 33  |  Issue : 3  |  Page : 164-167

Lumbar sympathetic block for pain relief and healing of chronic vascular ulcer on lower limb in patient with antiphospholipid syndrome and immune thrombocytopenic purpura


1 Pain Clinic, Department of Anesthesiology and Intensive Care, Hospital Taiping, Perak, Malaysia
2 Clinical Research Centre, Hospital Taiping, Ministry of Health, Perak, Malaysia

Date of Submission01-May-2019
Date of Decision25-Jun-2019
Date of Acceptance06-Aug-2019
Date of Web Publication5-Dec-2019

Correspondence Address:
Dr. Narwani Hussin
Clinical Research Centre, Hospital Taiping, Taiping 34000, Perak
Malaysia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpn.ijpn_33_19

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  Abstract 

Lower extremity ulcer is an infrequent but disabling complication of long-standing connective tissue disease such as antiphospholipid syndrome (APS). We reported a case of a 51-year-old female suffering from APS and immune thrombocytopenic purpura who was referred to our pain clinic with a history of 1-year chronic pain on the dorsal part of the bilateral foot. She also had a poor healing vascular ulcer on her right leg. Drug therapy managed to control her bilateral foot pain, but her chronic ulcer pain was not resolved, resulting in her decision to finally quit being a teacher. She even underwent 24 sessions of hyperbaric oxygen therapy but with unsatisfactory outcome. Later, a trial of lumbar sympathetic block was offered to her. Surprisingly, she responded very well. The chronic ulcer healed completely after her second session, and she was no longer dependent on any pain killer.

Keywords: Antiphospholipid syndrome, chronic leg ulcer, immune thrombocytopenic purpura, lumbar sympathetic block


How to cite this article:
Ghazali AI, Hussin N. Lumbar sympathetic block for pain relief and healing of chronic vascular ulcer on lower limb in patient with antiphospholipid syndrome and immune thrombocytopenic purpura. Indian J Pain 2019;33:164-7

How to cite this URL:
Ghazali AI, Hussin N. Lumbar sympathetic block for pain relief and healing of chronic vascular ulcer on lower limb in patient with antiphospholipid syndrome and immune thrombocytopenic purpura. Indian J Pain [serial online] 2019 [cited 2020 Feb 29];33:164-7. Available from: http://www.indianjpain.org/text.asp?2019/33/3/164/272378


  Introduction Top


Antiphospholipid syndrome (APS) is a thrombotic disorder defined by the presence of one or more clinical features of thrombosis and the presence of antiphospholipid antibodies (APLA).[1] APLAs are also frequently detected in immune thrombocytopenic purpura (ITP), a bleeding disorder, in which autoantibodies reacting to platelet-specific antigens; induce immune-mediated destruction of platelets.[2] One of the disabling complications of these diseases can be lower extremity ulcer.[3] The overall prevalence of chronic leg ulcer is approximately 1% in the United States, which is estimated to account for the financial burden of $2 billion/year.[4],[5] Despite the relatively small percentage of patients suffering from chronic leg ulcer, it has a significant impact on the patient quality of life such as painful experienced, impaired sleep, restricted mobility, and highly possible to affect and reduce the quality of work performance.[6],[7]

Treatment options for the ulcer vary from conservative management, medications, and surgical interventions.[8] The main aims of treatment are to facilitate the ulcer healing process and treat the pain. In this case report, we describe the resolution of medically refractory vascular ulcer of the lower limb in a patient with APS and ITP. This case is unusual and peculiar since she suffered from chronic leg ulcer caused by her underlying APS and ITP who was not responsive to the medical treatment used for APS and ITP and multiple sessions of hyperbaric oxygen therapy (HBOT). She was later successfully treated with lumbar sympathetic block (LSB).


  Case Report Top


A 51-year-old female who suffered from APS and ITP was referred to our pain clinic with the complaint of bilateral dorsal foot pain for more than a year. The pain was gradual in onset and worsened by prolonged walking. Further history revealed that she also had a chronic poor healing ulcer on her right calf, needing regular dressing that had also contributed to her chronic pain.

The patient was started on oral pregabalin 75 mg and oral celecoxib 200 mg once daily for her pain. At the same time, she was also on oral azathioprine 100 mg and oral hydroxychloroquine 200 mg once daily as prescribed by the medical team for her APS and ITP. Initially, the patient responded well to the medications. Her bilateral dorsal foot pain almost subsided after 3 months on the adjuvant drugs and pain killer. Nevertheless, there was no significant pain relief in her chronic vascular ulcer from the conservative management. She started to experience more frequent pain leading to sleep disturbance, which later on made her quit her profession as a teacher. There was no sign showing that the ulcer was going to heal. She was explained by the orthopedic team on the possibility of below-knee amputation if the condition became worse, but she refused it.

Later, we referred her for a trial of HBOT. Unfortunately, the result was not so promising even after 24 cycles [Figure 1], as the ulcer did not reduce in size, and her pain was still troublesome [Figure 2]. As a last resort, the patient was then offered a trial of LSB on which she agreed to. The procedure was done under fluoroscopy guidance at right L4 level using oblique tunnel view technique with the help of radioopaque contrast. A total of 6 ml of 0.375% ropivacaine together with 30 mg of triamcinolone was injected at the target area [Figure 3]. The first block had given excellent result where her chronic ulcer had shown great improvement with gradual reduction in the ulcer size, and she managed to cut down the drugs dosage to a minimal dose. Two months after the first block, her chronic ulcer had healed ≥80%. The initial ulcer size measured around 4 cm × 16 cm was reduced to a size around 1.5 cm × 8 cm only. We proceeded with the second LSB 10 weeks later, and the result was as excellent as the first one [Figure 4]. The ulcer was totally healed around 6 weeks after the second injection, and the patient was pain free after that [Figure 5]. Later, the patient was discharged pain free and without any pain medications from our pain clinic.
Figure 1: End position of the needle and contrast distribution

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Figure 2: The leg ulcer, (a) Before hyperbaric oxygen therapy, (b) After 24 cycles of hyperbaric oxygen therapy

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Figure 3: The leg ulcer 4 months after completed hyperbaric oxygen therapy and 2 days before the first lumbar sympathetic block on July 26, 2016

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Figure 4: The leg ulcer, (a) 2 months postlumbar sympathetic block, (b) 3 months postlumbar sympathetic block and 2 days before the second lumbar sympathetic block

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Figure 5: Around 6 weeks after second lumbar sympathetic block. The ulcer was almost healed. No more pain experienced by the patient

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  Discussion Top


The first interesting point to highlight about this case report was the fact that the chronic vascular ulcer on patient's right calf was not due to usual cause such as heavy tobacco use as in Buerger's disease (thromboangiitis obliterans). However, it was due to her underlying medical conditions which were APS and ITP. Lower extremity ulcers are late complication of connective tissue diseases and occur more commonly in patients with these diseases than in the general population. Although these lesions have historically been attributed to vasculitis, it is now recognized that inflammatory vessel injury accounts for fewer than 20% of ulcers in connective tissue disease.[3] The pathogenesis of these lesions is complex, and often, several processes act synergistically to initiate and perpetuate tissue injury. It has long been recognized that several factors may contribute to vessel occlusion, including abnormal vessel flow (due to the reduced ankle pump function in rheumatoid arthritis), abnormalities of the vessel wall or endothelium (due to inflammatory vasculitis or the vasculopathy associated with autoimmune diseases), and propensity of the blood to clot or inability to lyse clots when they occur.[9],[10],[11]

Second, it is worth to note that in this patient, despite being treated with hydroxychloroquine and azathioprine, the patient still suffered from chronic nonhealing ulcer. Literature show that hydroxychloroquine can reduce the risk of thrombosis in patients with APS, while azathioprine can act as an immunosuppressant therapy to decrease the titers of lupus anticoagulant and anticardiolipin antibodies.[12],[13] Another significant point to highlight was the patient had undergone multiple sessions of HBOT, a treatment in which the atmosphere is pressurized to three times higher than the normal atmospheric pressure, while 100% oxygen is being delivered. This will lead to more oxygen content being carried out by the patient's blood and help to stimulate the release of substances called growth factors and stem cells, which can promote healing.[14] Nevertheless, even with this expensive treatment, her chronic vascular ulcer remained in resistance condition.

The result in this case report showed that sympathetic block could be an option in treating patients with chronic vascular ulcer. According to the practice guidelines for chronic pain management by the American Society of Anesthesiologists, LSB are indicated for the treatment of multitude of sympathetic-mediated pain disorders.[15] Sympathetic nerve block works for resting pain and healing of ischemic ulcers because marked reduction in peripheral resistance leads to opening of arteriovenous anastomoses, thereby increasing blood flow to the skin.[16] Alleviation of resting pain also occurs because of the blockage of afferent pain fibers traveling in the sympathetic chain.[17]

Although the mechanism of how a mixture of a local anesthetic and a steroid medication can produce long-lasting pain relief is not completely understood,[18] it has been shown that steroids can block nociceptive input. Corticosteroids suppress discharges in chronic neuromas and prevent ectopic discharge in experimental neuromas, likely through a direct action on cell membrane.[19] The application of methylprednisolone has been shown to block C-fibers but not Aβ fibers.[20] Damaged nerve fibers often have a high accumulation of expressed sodium channels that are particularly sensitive to local anesthetics such as lidocaine.[21] Therefore, the sympathetic blocks with local anesthetics and steroids may provide pain relief through similar pharmacological actions on nociceptive fibers. Another study on chemical lumbar sympathectomy using phenol performed on 373 patients with ischemic lower limbs showed the successful result in 58.7% of the patients.[22]

As a conclusion, lumbar sympathectomy should be considered for patients with ischemic leg ulcer as an alternative to amputation in patients with otherwise viable limbs.

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.

Acknowledgment

The authors would like to thank the Director General of Health, Malaysia, for permission to publish this case report.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Wilson WA, Gharavi AE, Koike T, Lockshin MD, Branch DW, Piette JC, et al. International consensus statement on preliminary classification criteria for definite antiphospholipid syndrome: Report of an international workshop. Arthritis Rheum 1999;42:1309-11.  Back to cited text no. 1
    
2.
Galli M, Finazzi G, Barbui T. Thrombocytopenia in the antiphospholipid syndrome. Br J Haematol 1996;93:1-5.  Back to cited text no. 2
    
3.
Shanmugam VK, Steen VD, Cupps TR. Lower extremity ulcers in connective tissue disease. Isr Med Assoc J 2008;10:534-6.  Back to cited text no. 3
    
4.
Collins L, Seraj S. Diagnosis and treatment of venous ulcers. Am Fam Physician 2010;81:989-96.  Back to cited text no. 4
    
5.
Valencia IC, Falabella A, Kirsner RS, Eaglstein WH. Chronic venous insufficiency and venous leg ulceration. J Am Acad Dermatol 2001;44:401-21.  Back to cited text no. 5
    
6.
Ruckley CV. Socioeconomic impact of chronic venous insufficiency and leg ulcers. Angiology 1997;48:67-9.  Back to cited text no. 6
    
7.
Phillips T, Stanton B, Provan A, Lew R. A study of the impact of leg ulcers on quality of life: Financial, social, and psychologic implications. J Am Acad Dermatol 1994;31:49-53.  Back to cited text no. 7
    
8.
Robson MC, Cooper DM, Aslam R, Gould LJ, Harding KG, Margolis DJ, et al. Guidelines for the treatment of venous ulcers. Wound Repair Regen 2006;14:649-62.  Back to cited text no. 8
    
9.
Hafner J, Schneider E, Burg G, Cassina PC. Management of leg ulcers in patients with rheumatoid arthritis or systemic sclerosis: The importance of concomitant arterial and venous disease. J Vasc Surg 2000;32:322-9.  Back to cited text no. 9
    
10.
Pun YL, Barraclough DR, Muirden KD. Leg ulcers in rheumatoid arthritis. Med J Aust 1990;153:585-7.  Back to cited text no. 10
    
11.
Youssef P, Brama T, Englert H, Bertouch J. Limited scleroderma is associated with increased prevalence of macrovascular disease. J Rheumatol 1995;22:469-72.  Back to cited text no. 11
    
12.
Rand JH, Wu XX, Quinn AS, Chen PP, Hathcock JJ, Taatjes DJ, et al. Hydroxychloroquine directly reduces the binding of antiphospholipid antibody-beta2-glycoprotein I complexes to phospholipid bilayers. Blood 2008;112:1687-95.  Back to cited text no. 12
    
13.
Keswani SC, Chauhan N. Antiphospholipid syndrome. J R Soc Med 2002;95:336-42.  Back to cited text no. 13
    
14.
Löndahl M, Katzman P, Nilsson A, Hammarlund C. Hyperbaric oxygen therapy facilitates healing of chronic foot ulcers in patients with diabetes. Diabetes Care 2010;33:998-1003.  Back to cited text no. 14
    
15.
American Society of Anesthesiologists Task Force on Chronic Pain Management, American Society of Regional Anesthesia and Pain Medicine. Practice guidelines for chronic pain management: An updated report by the American Society of Anesthesiologists Task Force on Chronic Pain Management and the American Society of Regional Anesthesia and Pain Medicine. Anesthesiology 2010;112:810-33.  Back to cited text no. 15
    
16.
Agarwal P, Sharma D. Sympathectomy revisited: Current status in management of critical limb ischemia. In: Dieter RS, Dieter RA Jr., Dieter RA 3rd, Nanjundappa A, editors. Critical Limb Ischemia: Acute and Chronic. Switzerland: Springer International Publishing; 2016. p. 459-64.  Back to cited text no. 16
    
17.
Coventry BJ, Walsh JA. Cutaneous innervation in man before and after lumbar sympathectomy: Evidence for interruption of both sensory and vasomotor nerve fibres. ANZ J Surg 2003;73:14-8.  Back to cited text no. 17
    
18.
Van Boxem K, Cheng J, Patijn J, van Kleef M, Lataster A, Mekhail N, et al. 11. Lumbosacral radicular pain. Pain Pract 2010;10:339-58.  Back to cited text no. 18
    
19.
Devor M, Govrin-Lippmann R, Raber P. Corticosteroids suppress ectopic neural discharge originating in experimental neuromas. Pain 1985;22:127-37.  Back to cited text no. 19
    
20.
Johansson A, Bennett GJ. Effect of local methylprednisolone on pain in a nerve injury model. A pilot study. Reg Anesth 1997;22:59-65.  Back to cited text no. 20
    
21.
Craner MJ, Klein JP, Renganathan M, Black JA, Waxman SG. Changes of sodium channel expression in experimental painful diabetic neuropathy. Ann Neurol 2002;52:786-92.  Back to cited text no. 21
    
22.
Mashiah A, Soroker D, Pasik S, Mashiah T. Phenol lumbar sympathetic block in diabetic lower limb ischemia. J Cardiovasc Risk 1995;2:467-9.  Back to cited text no. 22
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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