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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 15  |  Issue : 4  |  Page : 374-377

Multisystem inflammatory syndrome in an adult associated with coronavirus disease-19 in a renal transplant recipient - A case report


Department of Nephrology and Transplantation, Institute of Kidney Diseases and Research Centre, Dr HL Trivedi Institute of Transplantation Sciences, Ahmedabad, Gujarat, India

Date of Submission14-May-2021
Date of Decision20-Jul-2021
Date of Acceptance29-Jul-2021
Date of Web Publication30-Dec-2021

Correspondence Address:
Dr. Hari Shankar Meshram
Department of Nephrology and Clinical Transplantation, Institute of Kidney Diseases and Research Centre, Dr HL Trivedi Institute of Transplantation Sciences, Ahmedabad, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijot.ijot_49_21

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  Abstract 

Coronavirus disease (COVID-19) caused by novel coronavirus (SARS-CoV-2) infection is still incompletely understood in transplantation, and there have been a few reports of multisystem inflammatory syndrome in adults (MIS-A) like illness in transplant patients. Herein, we report a case of MIS-A in a renal transplant that ultimately was successfully managed. The case was a 32-year-old man, transplanted 3 years ago, with chronic graft dysfunction and no other comorbidities. He presented with a 3-day history of fever and abdominal pain with no respiratory complaints. The patient had multi-organ involvement in the form of acute pancreatitis, severe diarrhea, acute kidney injury, and shock. Inflammatory markers including D-dimer and C-reactive protein were elevated. Chest radiology showed bilateral haziness on admission. The patient had two consecutive SARS CoV 2 reverse transcription–polymerase chain reaction (RT PCR) tests negative initially but eventually SARS CoV 2 antibody test came positive. The patient was managed initially with broad-spectrum antibiotics, and after confirmation of MIS-A, steroids, intravenous immunoglobulin, and anticoagulation were administered. The patient survived and was discharged on the 29th day of admission. Our reports highlight that MIS-A should be suspected in atypical cases irrespective of COVID-19 tests and should be confirmed with repeated RT-PCR and SARS-CoV-2 antibody tests.

Keywords: Coronavirus disease-19, multisystem inflammatory syndrome in adults, transplantation


How to cite this article:
Chauhan S, Meshram HS, Kute VB, Patel HV, Desai SN, Banerjee S, Dave RS, Deshmukh S. Multisystem inflammatory syndrome in an adult associated with coronavirus disease-19 in a renal transplant recipient - A case report. Indian J Transplant 2021;15:374-7

How to cite this URL:
Chauhan S, Meshram HS, Kute VB, Patel HV, Desai SN, Banerjee S, Dave RS, Deshmukh S. Multisystem inflammatory syndrome in an adult associated with coronavirus disease-19 in a renal transplant recipient - A case report. Indian J Transplant [serial online] 2021 [cited 2022 Jan 26];15:374-7. Available from: https://www.ijtonline.in/text.asp?2021/15/4/374/334433




  Introduction Top


Transplantation community is adversely affected in this pandemic with concerns of high mortality in recipients, halting of transplantation activities, donor safety, recipient safety, and transplantation from COVID-19 survivors.[1],[2],[3] In children, reports of multisystem inflammatory syndrome in children (MIS-C) is one such lethal and emerging aspect of COVID-19.[4],[5] The reports of MIS in adults (MIS-A) have been rarely described, and MIS-A in transplant population is reported rarely.[6] This entity poses both diagnostic and therapeutic dilemma and is encountered as a life-threatening condition in many cases. Herein, we report the first Indian case report of MIS-A in a renal transplant presenting to a high-volume transplant center of India. We hope this report will help the transplant clinicians in understanding this aspect of COVID-19 while dealing with critical care patients in intensive care units.


  Case Reports Top


A 32-year-old man received kidney transplant 3 years back with haplomatched brother and had chronic graft dysfunction with baseline serum creatinine of 2.5 mg/dl. His body mass index was 18.48 kg/m2. He was on triple immunosuppression consisting of steroids 5 mg, tacrolimus 0.06 mg/kg, and mycophenolic acid 360 mg BD dose. He had no family or contact history of COVID-19. He also had no other comorbidity and was apparently all right till October 2020, where he presented in our transplant outpatient department with 3 days of febrile illness associated with progressively worsening diffuse abdominal pain. He had no respiratory symptoms at presentation and his oxygen saturation was 98% on the day of admission. After admission on day 1, he developed multiple episodes (6–7 times per day) of nonbloody, watery, nonsticky, nonmucoid stools. On the 2nd day of admission, he developed hypotension requiring intravenous (IV) boluses and vasopressors (noradrenaline) and also developed difficulty of breathing with desaturation for which low-flow oxygen therapy was started. Physical examination was unremarkable except for diffuse abdominal tenderness on admission. His serum amylase was on admission 1650 IU/ml. CMV-DNA quantity and stool for cryptosporidium were negative. Blood and urine cultures were obtained on admission and were also negative. The patient had a high neutrophil–lymphocyte ratio of 6 on admission with peak of 11.2 on day 3. Other inflammatory markers such as D-dimer 4630 ng/ml and high-sensitive C-reactive protein (CRP) 120 mg/dl were also elevated. [Figure 1] shows the laboratory trends during the hospital course. The patient developed acute kidney injury (AKI) on admission, and complete recovery of AKI took 29 days. [Figure 2 demonstrates the timeline of the case. High-resolution computerized tomography thorax on the day of admission was suggestive of diffuse interstitial haziness predominately in the peripheral area typical of COVID-19. SARS-CoV-2 reverse transcription–polymerase chain reaction (RT-PCR) was done on the 2nd and 4th days, both of which came negative. SARS-CoV-2 IgG antibody test was done on the 6th day which was positive. He was started with empirical IV antibiotics (meropenem and levofloxacin). As per institutional management protocol, the patient's immunosuppression was altered as a stoppage of both tacrolimus and mycophenolic acid. Immunosuppression was reintroduced on the 14th day of admission as the condition improved. He received IV immunoglobulin (2 g/kg divided over 10 days), systemic glucocorticoid (IV methylprednisolone: 2 mg/kg/day for 10 days which was tapered and shifted to oral prednisolone over the next 2 weeks), and low-molecular-weight heparin (0.6 subcutaneous/day till discharge) for as a regimen for MIS-A. His pancreatitis symptoms were managed by bowel rest for 2 days and supportive care. Resolution of pancreatitis symptoms took around 6 days and severe diarrhea took around 8 days to settle. The patient was discharged home on the 29th day of hospital admission. His 6-month post-COVID follow-up is uneventful for any COVID-19-related sequelae.
Figure 1: Laboratory progress during the course of hospital stay. The monitored laboratory values were hemoglobin (g/dl) (neutrophil–lymphocyte ratio), serum creatinine (mg/dl), blood urea (mg/dl), and serum albumin (g/dl)

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Figure 2: Timeline of the renal transplant with multisystem inflammatory syndrome in adults from the onset of first symptoms to hospital course to discharge

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  Discussion Top


The impact of COVID-19 in transplantation is still an evolving process as substantial and high evidence-based data are accumulating with time from all parts of the world. MIS-C is a rare but severe complication of SARS-CoV-2 infection in children and adolescents.[7],[8],[9],[10] The reports of MIS-C were first highlighted with raising concerns in March 2020. The United States Centers for Disease Control and Prevention (CDC) case definition for MIS-C includes all of the three criteria: (1) any patient aged <21 years presenting with fever, raised inflammatory markers, and evidence of clinically severe illness mandating hospitalization with multi-organ involvement (>2); (2) no alternative plausible diagnoses other than COVID-19; and (3) positive for current or recent SARS-CoV-2 infection by RT-PCR, COVID-19 antibodies, or rapid antigen test. Moreover, in case COVID-19 tests are negative, a contact history is sufficient for classifying MIS-C with the above clinical scenario.[11] Subsequent to the reports of MIS-C, a similar illness was described in adults and is now recognized as MIS-A. As per the CDC, criteria for diagnosing MIS-A include (1) a severe illness mandating hospitalization in a person of age ≥21 years, (2) a positive test result for recent or past SARS-CoV-2 infection during admission or in the previous 12 weeks, (3) one or more extrapulmonary organ dysfunction (e.g., shock, cardiac dysfunction, arterial or venous thrombosis, pancreatitis, or acute liver injury), (4) laboratory evidence of severe inflammation (e.g., elevated CRP and D-dimer), and (5) the absence of severe respiratory illness. Our case fulfilled all the criteria of MIS-A. Although MIS-A-like illness has been described in literature in general population,[12] there had been no reports in transplant patients. To the best of our knowledge, this is the first case report of MIS-A in a renal transplant patient from India. The patient was apparently alright and then developed multi-organ involvement and systemic inflammation leading to critical illness needing intensive care management. Of note, the delay in diagnosis was due to the absence of respiratory complaints on admission. It is relatively clear now that SARS-CoV-2 can involve any organs including the brain, heart, liver, kidneys, and gastrointestinal tract other than the respiratory tract.[13],[14] The interval between SARS-CoV-2 infection and the development of MIS-A is still to be known. Our case had no history of any symptoms of mild COVID-19 in the recent past, and so if such cases present with MIS, the diagnosis becomes more difficult. The immune pathogenesis of MIS-A is enigmatic and in transplant settings is further blurred, but it is speculated to be a postinfectious sequela of acute COVID-19 rather than acute COVID-19 itself.[15] The potential underlying mechanism includes endothelial damage, thrombosis, inflammation, dysregulated immunity, and renin–angiotensin–aldosterone dysregulation. The case had multi-organ involvement including GI involvement in the form of severe diarrhea, severe abdominal pain, pancreatitis, cardiac involvement in the form of hypotension and low voltage complexes, and renal involvement in the form of AKI. The complexity in managing the case was in diagnosing the case as COVID-19 as repeated RT-PCR was negative despite clinical scenario, chest imaging, and blood reports simulating COVID-19. Another important highlight is the decision of the usage of steroids in this case, which could have been detrimental if the case was not due to COVID-19. COVID-19 antibody tests helped in making the correct diagnosis and hence emphasize the need of combined antibody and RT-PCR testing, especially in dicey clinical scenarios. There is no specific therapy for COVID 19 and guidelines are still getting upgraded. In our case, the patient survived fortunately with IV steroids, IV immunoglobulin, anticoagulation, and oxygen support. A very important and differentiating feature of MIS from severe COVID 19 is the absent or mild respiratory involvement in the former. This is not surprising that no reports of MIS-A in transplant patients are published owing to the nature of MIS-A as many cases would have remained unnoticed. The true burden of MIS-A in transplant settings cannot be commented upon. Further research is needed to explore the pathogenesis and clinical spectrum of MIS-A in transplant patients.


  Conclusion Top


MIS-A in transplant patients is a serious and life-threatening condition and a multidisciplinary approach to treatment is necessitated for management of the patient, as the condition can potentially involve any organ. The prime learning point from the report is the fact that in cases of critical patients with multi-organ involvement, MIS-A linked to COVID-19 should be suspected and confirmed with antibody tests when COVID-19 RT-PCR is negative.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Kute VB, Bhalla AK, Guleria S, Ray DS, Bahadur MM, Shingare A, et al. Clinical profile and outcome of COVID-19 in 250 kidney transplant recipients: A multicenter cohort study from India. Transplantation 2021;105:851-60.  Back to cited text no. 1
    
2.
Raja MA, Mendoza MA, Villavicencio A, Anjan S, Reynolds JM, Kittipibul V, et al. COVID-19 in solid organ transplant recipients: A systematic review and meta-analysis of current literature. Transplant Rev (Orlando) 2021;35:100588.  Back to cited text no. 2
    
3.
Kute VB, Godara S, Guleria S, Ray DS, Aziz F, Hegde U, et al. Is it safe to be transplanted from living donors who recovered from COVID-19? experience of 31 kidney transplants in a multicenter cohort study from India. Transplantation 2021;105:842-50.  Back to cited text no. 3
    
4.
Greene AG, Saleh M, Roseman E, Sinert R. Toxic shock-like syndrome and COVID-19: Multisystem inflammatory syndrome in children (MIS-C). Am J Emerg Med 2020;38:2492.e5-6.  Back to cited text no. 4
    
5.
Godfred-Cato S, Bryant B, Leung J, Oster ME, Conklin L, Abrams J, et al. COVID-19–associated multisystem inflammatory syndrome in children–United States, March–July 2020. MMWR Morb Mortal Wkly Rep 2020;69:1074.  Back to cited text no. 5
    
6.
Chavarot N, Burger C, Aguilar C, Scemla A, Sberro-Soussan R, Amrouche L, et al. Ig-responsive relapsing inflammatory syndrome following COVID-19 in a kidney transplant recipient. Kidney Int 2021;99:767-8.  Back to cited text no. 6
    
7.
Dufort EM, Koumans EH, Chow EJ, Rosenthal EM, Muse A, Rowlands J, et al. Multisystem inflammatory syndrome in children in New York State. N Engl J Med 2020;383:347-58.  Back to cited text no. 7
    
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Kofman AD, Sizemore EK, Detelich JF, Albrecht B, Piantadosi AL. A young adult with COVID-19 and multisystem inflammatory syndrome in children (MIS-C)-like illness: A case report. BMC Infect Dis 2020;20:716.  Back to cited text no. 8
    
9.
Dolinger MT, Person H, Smith R, Jarchin L, Pittman N, Dubinsky MC, et al. Pediatric Crohn's disease and multisystem inflammatory syndrome in children (MIS-C) and COVID-19 treated with infliximab. J Pediatr Gastroenterol Nutr 2020;71:153-5.  Back to cited text no. 9
    
10.
Feldstein LR, Rose EB, Horwitz SM, Collins JP, Newhams MM, Son MB, et al. Multisystem inflammatory syndrome in U.S. children and adolescents. N Engl J Med 2020;383:334-46.  Back to cited text no. 10
    
11.
Centers for Disease Control and Prevention: Multisystem Inflammatory Syndrome in Children (MIS-C) Associated with Coronavirus Disease 2019 (COVID-19); 2020. Available from: https://emergency.cdc.gov/han/2020/han00432.asp?deliveryName=USCDC_511-DM28431. [Last accessed on 2021 Apr 10].  Back to cited text no. 11
    
12.
Hékimian G, Kerneis M, Zeitouni M, Cohen-Aubart F, Chommeloux J, Bréchot N, et al. Coronavirus disease 2019 acute myocarditis and multisystem inflammatory syndrome in adult intensive and cardiac care units. Chest 2021;159:657-62.  Back to cited text no. 12
    
13.
Gupta A, Madhavan MV, Sehgal K, Nair N, Mahajan S, Sehrawat TS, et al. Extrapulmonary manifestations of COVID-19. Nat Med 2020;26:1017-32.  Back to cited text no. 13
    
14.
Shaigany S, Gnirke M, Guttmann A, Chong H, Meehan S, Raabe V, et al. An adult with Kawasaki-like multisystem inflammatory syndrome associated with COVID-19. Lancet 2020;396:e8-10.  Back to cited text no. 14
    
15.
Morris SB, Schwartz NG, Patel P, Abbo L, Beauchamps L, Balan S, et al. Case series of multisystem infammatory syndrome in adults associated with SARS-CoV-2 infection–United Kingdom and United States, March–August 2020. MMWR Morb Mortal Wkly Rep 2020;69:1450-6.  Back to cited text no. 15
    


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