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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 9  |  Issue : 1  |  Page : 43-47

Complex regional pain syndrome (reflex sympathetic dystrophy) after coronary artery bypass graft – An interesting case


Senior Consultant in Internal Medicine and Diabetes, Prem Health Care, Mysore, Karnataka, India

Date of Submission01-Dec-2019
Date of Decision08-Feb-2020
Date of Acceptance09-Feb-2020
Date of Web Publication03-Feb-2021

Correspondence Address:
Dr. Manjunath Premanath
Prem Health Care, 671, Nrupatunga Road, M-block, Kuvempunagar, Mysore - 570 023, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/AJIM.AJIM_81_19

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  Abstract 


A 58-year-old male presented himself with swelling, pain, and color change of the left shoulder, arm, and hand of 3 months duration. His problems started a week after he underwent a coronary artery bypass graft for triple-vessel coronary artery disease. The function of the left upper limb was severely restricted. Investigations done to know the cause of his problems were negative. A clinical diagnosis of complex regional pain syndrome was made. Even though this syndrome was observed after myocardial infarction, its occurrence after CABG has been quite rare. This case is presented as CABG is on the rise, and more and more cases may occur which should not be missed.

Keywords: Complex regional pain syndrome, coronary artery bypass graft, reflex sympathetic dystrophy


How to cite this article:
Premanath M. Complex regional pain syndrome (reflex sympathetic dystrophy) after coronary artery bypass graft – An interesting case. APIK J Int Med 2021;9:43-7

How to cite this URL:
Premanath M. Complex regional pain syndrome (reflex sympathetic dystrophy) after coronary artery bypass graft – An interesting case. APIK J Int Med [serial online] 2021 [cited 2021 Feb 28];9:43-7. Available from: https://www.ajim.in/text.asp?2021/9/1/43/308654




  Introduction Top


Complex regional pain syndrome (CRPS) is a disorder of extremities, with autonomic and vasomotor instability, albeit not very common, the diagnosis of which is usually missed. It was also called reflex sympathetic dystrophy (RSD) and shoulder-hand syndrome (SHS) earlier but not anymore.[1] Since the primary role of the autonomic nervous system could not be implicated in the pathogenesis, the term RSD was changed to CRPS.[2] CRPS is a term used for quite a few disorders characterized by spontaneous or stimulus-induced pain, which is disproportionate to the event that precipitated it.[3] Diagnosis is purely clinical after ruling out other causes, as the symptoms cannot be explained by the initial trauma.[4] SHS, as it was called earlier, was known to occur after myocardial infarction.[1] This case occurred after a coronary artery bypass graft (CABG) and the diagnosis was missed. Since a good number of CABG's are being done every day, clinicians have to be aware of the occurrence of this syndrome, as the early diagnosis may help the patient to a certain extent.


  Case Report Top


Mr, B a 58 years old male complained of pain and swelling of left arm, forearm and swelling and difficulty to move the fingers of the left hand for the last 3 months.

The patient noticed swelling and pain in the left arm, forearm, and hand a week after he underwent coronary artery bypass surgery (CABG) for triple-vessel coronary artery disease. He had difficulty to grip the objects in his left hand. He noticed discoloration of the skin of the affected hand also [Figure 1],[Figure 2],[Figure 3],[Figure 4]. The disability continued to increase for the past 3 months. He was prescribed anti-inflammatory analgesics, which were of no avail.
Figure 1: Swelling of the left shoulder

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Figure 2: Swelling of the left hand

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Figure 3: Unable to flex left fingers

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Figure 4: Color changes on the dorsum of the left hand

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Clinical examination revealed an elderly male, moderately nourished with swelling of the left shoulder, arm forearm, and hand. Touching the area was extremely painful. The skin was warm to touch. There was swelling of the smaller joints of the hand and had difficulty in flexing the fingers. There was discoloration on the dorsum of the hand. The movements at the shoulder and elbow were also painful. Systemic examination was normal. The patient was not a diabetic or smoker.

The patient was a known case of ischemic heart disease and had inferior wall myocardial infarction a few years ago [Figure 5]. He was an agriculturist with poor socioeconomic background. He was admitted with acute chest pain to a tertiary hospital in May 2019, where a diagnosis of acute coronary syndrome was made, and a coronary angiogram was done, which showed the left anterior descending artery (LAD) type 3 vessel, proximal LAD 90% lesion with calcification, left circumflex artery, nondominant with proximal 90% short-segment lesion, obtuse marginal artery (OMA) 2.5–3 mm vessel with ostioproximal 50% lesion, and right coronary artery, a dominant vessel, small caliber, diffuse long-segment lesion with 90% obstruction [Figure 6]. Other investigations done at that time such as hemoglobin percentage, serum electrolytes, HIV I and II, HbS Ag, and tests for syphilis were all normal. Echocardiography (ECHO) showed left ventricular segment hypokinesia, ejection fraction (EF) 43% with trivial mitral regurgitation.
Figure 5: Electrocardiography showing old inferior wall myocardial infarction

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Figure 6: Coronary angiography

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The patient was admitted for CABG 1 month later, and his investigations such as lipid profile, random blood sugar, Troponin T, creatine phosphokinase-MB (CPK-MB), thyroid-stimulating hormone, liver function tests, serum electrolytes, and erythrocyte sedimentation rate (ESR) were within the normal limits. X-ray of the chest was normal [Figure 7] ECHO showed diastolic dysfunction with EF of 60%. Computed tomography carotid and cerebral angiogram showed some soft plaques with calcification to the tune of 50%–70% in the left and right common carotid artery bulb area. He underwent CABG with two grafts, one off-pump left internal mammary artery to LAD and the other on-pump beating heart, saphenous venous graft to OMA. Postsurgery his Troponin T and CPK-MB values were high for 3 days and settled to normal later. He was discharged after 6 days of stay in the hospital in a stable condition with advice to take statins, clopidogrel, aspirin, beta-blockers, and ivabradine.

A week after surgery, he started having pain in the left upper limb, he went to the hospital again, where arterial Doppler study of the left upper limb arteries was done, which was normal [Figure 8]. X-ray of the cervical spine AP and lateral was done, which was also normal [Figure 9]. At the present visit, his white blood cells total count and differential count, ESR, rheumatoid arthritis factor, and antinuclear antibody were done and were within the normal limits.
Figure 7: X-ray chest

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Figure 8: X-ray cervical spine

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Figure 9: Doppler of the left upper limb arteries

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A clinical diagnosis of CRPS was made as a process of exclusion of other causes.


  Discussion Top


CRPS develops usually after the surgery or physical trauma and is characterized by vasomotor dysfunction, pseudo motor abnormality, and focal edema either alone or in combination. The syndrome is divided into two types: CRPS type 1 and CRPS type 2.[2]

CRPS type 1 is seen in some patients after tissue injury, myocardial infarction, or stroke. Pain is localized to one arm or leg with edema, which is a constant feature. Alteration in the color of the skin, swelling of the smaller joints with restriction of movements, alteration in the temperature of the skin, and dystrophic changes are also seen. What has to be borne in mind is that the severity of symptoms is not related to the severity of the preceding trauma, and the findings are not confined to a single nerve distribution.[1],[2],[5]

CRPS type 2 develops after an injury to a specific nerve with other symptoms and signs being common.[2] CRPS has three phases once it starts. In the first phase, pain and swelling start in the distal extremities in a week to 3 months after the precipitating event. Pain would be severe, throbbing or ache, with warm edematous extremity and limitation of movements. In the second phase which is from 3 to 6 months of time, pseudo motor symptoms appear with the affected limb becoming cooler with shiny skin, and in the third phase which would be after 6 months, trophic changes in the nails and skin start to appear with flexion contractures.[2]

Why would all these occur? It was thought earlier that there was a cross-connection between the efferent sympathetic and afferent somatic pain fibers. When it was shown that the pain could be abolished by the depletion of neurotransmitters, it was confirmed that the cross-excitation was chemical and not electrical. The present explanation for the pain that occurs in CRPS is that of an abnormal adrenergic sensitivity which develops in the injured nociceptors, and the locally circulating neurotransmitters trigger a painful response because of this heightened sensitivity.[6] The molecular changes that occur in the sensory neurons, as well as the spinal cord, lead to alteration in N-Methyl-D Aspartate receptors with the induction of cyclooxygenase and prostaglandin synthesis. Changes have been observed in the GABAergic inhibition in the dorsal horns.[6] Recently, parietal lobe dysfunction with problems in sensory and motor integration with abnormality in the immune system is also shown. The clinical picture of this syndrome has been explained by the presence of inflammation, interaction between the immune system and autonomic nervous system with aberrant neuroplasticity.[3]

Coming to the present case, this patient developed symptoms in his left shoulder and upper limb a week after CABG which progressed. Other cervicobrachial syndromes and thoracic out let obstruction and vascular lesions have been thought earlier and had been ruled out by relevant investigations such as X-ray of the cervical spine and upper limb Doppler. The investigations done later to rule out rheumatoid arthritis or any other autoimmune disease were also negative. Analgesics and other methods to relieve pain earlier were ineffective. Since the diagnosis is purely clinical[7],[8] and by a process of exclusion of other conditions, a diagnosis of CRPS was made. CRPS occurring after myocardial infarction is very well known, but that occurring after a procedure of CABG is not commonly reported. One case report has been published of CRPS occurring after CABG, and they claimed that theirs was the first case reported.[9]

The treatment of CRPS is not satisfactory.[10] Routine analgesics do not help in relieving the pain. Gabapentin and pregabalin have been used with mixed results. A short course of steroids has been found to be beneficial. Early rehabilitation with physiotherapy may prevent flexor contractures.[2] This patient had routine analgesics and pregabalin, which did not reduce his pain. A short course of steroids was given, and the patient found a reduction in pain and swelling.

CABG is a very common surgery nowadays, and clinician has to be aware that CRPS can occur after CABG. Many cases might have been missed because of unawareness on the part of the clinician. Early diagnosis and management may help the patient to a certain extent in avoiding disability.

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.

Addendum to Treatment

This patient came for follow-up after 10 months. A course of steroids had done wonders to him. His swelling of the left shoulder left hand and fingers had completely come down. Movements of both left shoulder and left hand had become normal [Figure 10] and [Figure 11]. This showed that, while the results of the treatment usually is unsatisfactory, complete recovery as happened in this case gives hope for patients of CRPS.
Figure 10: Dorsum of the hand

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Figure 11: Flexed fingers

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  References Top

1.
Hellman DB, Imboden JB Jr. Rheumatologic, immunologic and allergic disorders in current medical diagnosis and treatment. Ed: Maxine A, Papadakis, McPhee SJ. Lange. 57th ed. Ch. 20. USA: McGraw Hill; 2018. p. 883.  Back to cited text no. 1
    
2.
Low PA, Engshom JW. Disorders of the autonomic nervous system in Harrison's principles of internal medicine. Ed: Kasper DL, Hauser SL, Jameson JL, Fauci AS, Longo DL, Loscalzo J. 19th ed. Ch. 454, Vol. 2.USA: McGraw Hill; 2015. p. 2643-44.  Back to cited text no. 2
    
3.
Borchers AT, Gershwin ME. Complex regional pain syndrome: A comprehensive and critical review. Autoimmun Rev 2014;13:242-65.  Back to cited text no. 3
    
4.
Birklein F, O'Neill D, Schlereth T. Complex regional pain syndrome: An optimistic perspective. Neurology 2015;84:89-96.  Back to cited text no. 4
    
5.
Urits I, Shen AH, Jones MR, Viswanath O, Kaye AD. Complex regional pain syndrome, current concepts and treatment options. Curr Pain Headache Rep 2018;22:10.  Back to cited text no. 5
    
6.
Ropper AH, Samuels MA, editors. Causalgia and Reflex Sympathetic Dystrophy (Complex Regional Pain Syndrome) in Adams and Victor's Principles of Neurology. 9th ed. Sec. 2, Ch. 8. USA: McGraw Hill Medical; 2009. p. 137-8.  Back to cited text no. 6
    
7.
Rockett M. Diagnosis, mechanisms and treatment of complex regional pain syndrome. Curr Opin Anaesthesiol 2014;27:494-500.  Back to cited text no. 7
    
8.
Brunner F. Complex regional pain syndrome. Z Rheumatol 2017;76:335-47.  Back to cited text no. 8
    
9.
Ediz L, Ceylan MF, Hiz O, Tekeoglu I. Complex regional pain syndrome type 1 (shoulder hand syndrome) in an elderly patient after open cardiac surgical intervention: A case report. Eastern J Med 2011;16:56-8.  Back to cited text no. 9
    
10.
Steinbrocker O, Argyros TG. The shoulder-hand syndrome: Present status as a diagnostic and therapeutic entity. Med Clin North Am 1958;42:1533-53.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]



 

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