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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 31  |  Issue : 3  |  Page : 191-193

An atypical case of postsurgical complex regional pain syndrome in a patient having nonhealing varicose venous ulcer treated by lumbar sympathectomy


1 Department of Pain Management, Pain Clinic of Pvt. Ltd; Detartment of Anesthesia and Pain Management – King Edward Memorial (KEM) Hospital, Mumbai, Maharashtra, India
2 Fellow of Pain Medicine, Pain Clinic of Pvt Ltd, Mumbai, Maharashtra, India

Date of Web Publication18-Jan-2018

Correspondence Address:
Kailash Kothari
2005/A, Cosmic Heights Bhakti Park, Wadala, Mumbai - 400 037, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpn.ijpn_80_17

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  Abstract 


Complex regional pain syndrome (CRPS) of the lower limb is a relatively uncommon entity as compared to CRPS of the upper extremity. Literature search has revealed only 2 retrospective case series and a single case report of lower extremity CRPS type I from 1975 to 2014 on Pubmed, isolated cases of CRPS type I of lower extremity have also been reported following knee surgeries and arthroscopies. This report presents a case of lower limb CRPS type I, following surgery for varicose vein ulcer. Pain was not relieved with medications. Diagnostic lumbar sympathectomy was done and patient had tremendous relief of pain following that, proving sympathetic mediated pain of the involved limb.


How to cite this article:
Kothari K, Garg A, Vignesh S, Patel B, Tilvawala K. An atypical case of postsurgical complex regional pain syndrome in a patient having nonhealing varicose venous ulcer treated by lumbar sympathectomy. Indian J Pain 2017;31:191-3

How to cite this URL:
Kothari K, Garg A, Vignesh S, Patel B, Tilvawala K. An atypical case of postsurgical complex regional pain syndrome in a patient having nonhealing varicose venous ulcer treated by lumbar sympathectomy. Indian J Pain [serial online] 2017 [cited 2019 Sep 20];31:191-3. Available from: http://www.indianjpain.org/text.asp?2017/31/3/191/223672




  Introduction Top


Complex regional pain syndrome (CRPS) is a complex and poorly understood condition which was known previously by varying names, most commonly by the term reflex sympathetic dystrophy and causalgia.[1] Specific diagnostic criteria were given by the international association for the study of pain (IASP) as depicted in [Table 1].[2] CRPS may affect either upper or lower limb, may be localized, or may involve the whole extremity. Rarely, it affects >1 limb.[3] CRPS involving the lower extremity is a diagnostic and a therapeutic challenge to the physicians as it is an uncommon disease with a prevalence of <2% in most retrospective series.[4] A study from the Netherlands reported an incidence of 26.2 cases per 100,000 person-years, whereas a study from the United States estimated the incidence at 5.5 cases per 100,000 person-years.[5]
Table 1: Budapest CRPS diagnostic criteria

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The success of various treatment modalities differs in the upper and lower limb as lower limbs are more refractory to treatment.[6] This differential response has led some to recognize the CRPS of lower limbs as a distinct clinical entity.[7]

Probably, there is no case report showing CRPS in postsurgical varicose venous ulcer.

Usually, the surgery done for varicose veins relieves this pain. In our case, the pain did not get relieved, rather increased disproportionately over the past 5 years. The patient developed clinical findings suggestive of CRPS Type I, postsurgery, refractory to medical management. In such cases, lumbar sympathetic nerve block is used to diagnose and treat sympathetically mediated pain.


  Case Report Top


A 52-year-old male presented with a history of varicose veins in the left leg since 7 years. He developed ulcer in left lower limb 5 years back. Hence, he got operated for varicose veins 5 years back. Stripping of the varicose veins was done. Following surgery, the ulcer did not heal. The patient started developing pain in that area.

Pain was severe, excruciating, burning in nature. There was pain even with nonpainful stimuli such as touch or blowing over the skin. The severity of the pain was graded by visual analog scale (VAS). It was found out to be 8/10. Pain appeared even at rest and worsened when walking and standing for prolonged duration. On examination, the patient had local tenderness on the area around the ulcer.

Sensory examination in the affected extremity revealed hyperalgesia around the margins of the ulcer. The range of motion was normal at the ankle joint. Skin around the ulcer was shiny, edematous with normal pulsation. The swelling extended up to the foot [Figure 1]a.
Figure 1: a) Non healing ulcer; b) Healing ulcer post procedure. Note that the patient's pain was relieved completely but ulcer didn't heal completely so he was referred to the vascular surgeon for varicose vein management

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He tried various medications for the pain. The patient had taken tramadol, gabapentin, and amitriptyline for pain but there was not much relief. Ulcer also turned out into a nonhealing ulcer.

The patient was also advised for the vascular surgery for the nonhealing ulcer. However, the patient did not want surgery because of the bad experience of the previous surgery. Hence, he decided to undergo diagnostic sympathectomy.

Due to refractory pain, diagnostic lumbar sympathectomy block was planned. He was extensively investigated elsewhere, but his hematology and biochemistry investigations reports were normal.

Written and informed consent for the procedure was taken from the patient.

After admission to the operating room, monitors were applied to the patient-electrocardiogram, pulse oximetry, and noninvasive blood pressure. Intravenous cannula was taken and preloading with adequate amount of fluid was done. The patient was positioned in prone position. The patient was maintained on spontaneous respiration with O2 supplementation through nasal catheter.

Painting and draping of lower back area was done under all aseptic and antiseptic precautions. Local anesthesia was given in skin and subcutaneous tissue with lignocaine 2%. The blockade was performed with the aid of fluoroscopy, on the left side at the L2 and L4 level, both with 15 cm 22G Quincke needle. Following confirmation of needle positioning and observation of contrast dispersion at each level, 7.5 ml 0.25% bupivacaine was given at each level [Figure 2]. The patient was observed after that. The patient had complete pain relief following the procedure. Hence, it was confirmed that pain of the patient was sympathetically mediated.
Figure 2: Fluoroscopy guided left-sided lumbar sympathectomy

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We had planned radiofrequency neurolysis of left side lumbar sympathetic chain if pain recurs. However, the patient had tremendous relief only with the diagnostic block. His VAS score came down to 2/10. Hence, we had not given therapeutic block. His ulcer also started healing after the block as shown in [Figure 1]b. Patient has good pain relief at the end of 6 months from the date of procedure. Varicose veins persisted, so he was referred to vascular surgeon for the definitive treatment for the varicose vein.


  Discussion Top


We had an unusual case of varicose ulcer patient where pain did not get relieved even after getting operated for varicose veins. The presence of nonhealing ulcer masks the clinical features of CRPS as edema, skin change color and pain around ulcer is thought to be due to the ulcer. However, there should always be a high suspicion of CRPS in such patients.

Treatment of CRPS has not yet achieved precise standard guidelines and doctors, as well as patients, face difficulties with the diagnosis and management. Treatment should be started as early as possible to avoid secondary complications of chronicity of pain and fear, anxiety of living with undiagnosed pain. Because of the variation in treatment approach, an integrated interdisciplinary treatment approach is recommended, tailored to the individual patient.[8]

CRPS has a variable presentation. Nearly 95% cases of CRPS have a history of trauma or surgery.[9] Most commonly within the first 6 weeks, due to the sympathetic overreaction, there is a swollen, immobile, and painful limb. Pain does not follow the distribution of a peripheral nerve and is often described as burning in nature. There is increased sweating, with color changes in the extremity in most of the patients. Frequently, there is allodynia, i.e., pain elicited with a nonnoxious normal stimulus such as light touch of a bed sheet and blowing of air. Late in the course of the disease, there are trophic changes (sudomotor): dystrophic, smooth, shiny skin; osteoporosis; fast growing and brittle nails; hypertrichosis; and muscular and subcutaneous atrophy. All these symptoms may be found along with joint swelling and contractures.[10]

In our case, the patient had disproportionate pain to stimuli, hyperalgesia, skin color changes, and swelling around the ulcer area. Hence, we diagnosed our patient with CRPS as his clinical features fit into IASP diagnostic criteria for CRPS.

Akkoc et al. reported a case with bilateral lower extremity CRPS in a patient with paraplegia occurring following spinal disc herniation surgery, who was treated with anticonvulsants and opioids for 6 months. She remained well but had a recurrence of symptoms for which she was treated successfully with pulsed radiofrequency (PRF) lumbar sympatholysis.[11]

Saranita et al. reported a case of CRPS who developed the same after ankle injury and surgery highlighting the treatment options for the same including spinal cord stimulation (SCS). They reported the clinical evidence of SCS to be of level 4.[12]

Manjunath et al. reported 20 cases of lower extremity CRPS and compared the efficacy of percutaneous radiofrequency lumbar sympathectomy and phenol lumbar sympathetic neurolysis and found the two to be comparable.[13]

Newer treatments like intrathecal baclofen, PRF along with SCS have been shown to have some evidence in literature.[14]

There are very few case reports where CRPS of lower limb got treated only by diagnostic lumbar sympathectomy, and therapeutic block was not needed.

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.
Boas R. Complex regional pain syndromes: Symptoms, signs and differential diagnosis. In: Janig W, Stanton-Hicks M, editors. Reflex Sympathetic Dystrophy: A Reappraisal. Seattle, WA: IASP Press; 1996. p. 79-92.  Back to cited text no. 1
    
2.
Merskey H, Bogduk N. Classification of Chronic Pain: Descriptions of Chronic Pain Syndromes and Definitions of Pain Terms. Seattle, WA: IASP Press; 1994.  Back to cited text no. 2
    
3.
Schiffenbauer J, Fagien M. Reflex sympathetic dystrophy involving multiple extremities. J Rheumatol 1993;20:165-9.  Back to cited text no. 3
    
4.
Albazaz R, Wong YT, Homer-Vanniasinkam S. Complex regional pain syndrome: A review. Ann Vasc Surg 2008;22:297-306.  Back to cited text no. 4
    
5.
de Mos M, de Bruijn AG, Huygen FJ, Dieleman JP, Stricker BH, Sturkenboom MC, et al. The incidence of complex regional pain syndrome: A population-based study. Pain 2007;129:12-20.  Back to cited text no. 5
    
6.
Wang JK, Johnson KA, Ilstrup DM. Sympathetic blocks for reflex sympathetic dystrophy. Pain 1985;23:13-7.  Back to cited text no. 6
    
7.
Katz MM, Hungerford DS. Reflex sympathetic dystrophy affecting the knee. J Bone Joint Surg Br 1987;69:797-803.  Back to cited text no. 7
    
8.
Bruehl S, Harden RN, Galer BS, Saltz S, Bertram M, Backonja M, et al. External validation of IASP diagnostic criteria for complex regional pain syndrome and proposed research diagnostic criteria. International association for the study of pain. Pain 1999;81:147-54.  Back to cited text no. 8
    
9.
Bruehl S, Harden RN, Sorrel P. Complex regional pain syndromes, a fresh look at a difficult problem. 62nd Annual Assembly of the American Academy of Physical Medicine and Rehabilitation. Conference Proceedings. San Francisco; 2000.  Back to cited text no. 9
    
10.
Lindenfeld TN, Bach BR Jr., Wojtys EM. Reflex sympathetic dystrophy and pain dysfunction in the lower extremity. Instr Course Lect 1997;46:261-8.  Back to cited text no. 10
    
11.
Akkoc Y, Uyar M, Oncu J, Ozcan Z, Durmaz B. Complex regional pain syndrome in a patient with spinal cord injury: Management with pulsed radiofrequency lumbar sympatholysis. Spinal Cord 2008;46:82-4.  Back to cited text no. 11
    
12.
Saranita J, Childs D, Saranita AD. Spinal cord stimulation in the treatment of complex regional pain syndrome (CRPS) of the lower extremity: A case report. J Foot Ankle Surg 2009;48:52-5.  Back to cited text no. 12
    
13.
Manjunath PS, Jayalakshmi TS, Dureja GP, Prevost AT. Management of lower limb complex regional pain syndrome type 1: An evaluation of percutaneous radiofrequency thermal lumbar sympathectomy versus phenol lumbar sympathetic neurolysis – A pilot study. Anesth Analg 2008;106:647-9.  Back to cited text no. 13
    
14.
Zuniga RE, Perera S, Abram SE. Intrathecal baclofen: A useful agent in the treatment of well-established complex regional pain syndrome. Reg Anesth Pain Med 2002;27:90-3.  Back to cited text no. 14
    


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