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Inflammatory Bowel Disease Presenting with Concurrent COVID-19 Multisystem Inflammatory Syndrome Katherine F

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Inflammatory Bowel Disease Presenting with Concurrent COVID-19 Multisystem Inflammatory Syndrome Katherine F Inflammatory Bowel Disease Presenting With Concurrent COVID-19 Multisystem Inflammatory Syndrome Katherine F. Sweeny, MD,a,p Yanjia J. Zhang, MD, PhD,a,p Bonnie Crume, MD,b Colin A. Martz, MD, PhD,b Melissa M. Blessing, DO,c Stacy A. Kahn, MDa Coronavirus disease 2019 is associated with a postinfectious multisystem abstract inflammatory syndrome in children (MIS-C). This syndrome is marked by cytokine storm and multiorgan dysfunction, often affecting the gastrointestinal tract, the heart, and the hematopoietic system. We describe the case of a 16-year-old boy with an initial presentation of severe inflammatory bowel disease and concurrent MIS-C. He presented with abdominal pain, diarrhea, and hematochezia and met criteria for the systemic inflammatory response syndrome. Laboratory inflammatory profiling aDivision of Gastroenterology, Hepatology and Nutrition and bDepartments of Medicine, and cPathology, Boston revealed markedly elevated ferritin, D-dimer, C-reactive protein, soluble Children’s Hospital, Boston, Massachusetts interleukin 2, and interleukin 6 levels. Endoscopy and colonoscopy revealed pContributed equally as co-first authors severe active gastroduodenitis, patchy colitis, and a normal-appearing terminal ileum. The patient was treated with a combination of steroids, Drs Zhang, Sweeny, and Kahn conceptualized the report and drafted the initial manuscript; Drs Crume intravenous immunoglobulin, and infliximab, and his symptoms slowly and Martz collected patient data and analyzed data resolved over a 3-week period. In this case, we describe coincident MIS-C with trends; Dr Blessing collected and analyzed a remarkably severe and difficult-to-treat initial presentation of inflammatory histopathologic data; and all authors reviewed and revised the manuscript and approved the final bowel disease and highlight the need to investigate the effect of coronavirus manuscript as submitted. fl disease 2019 and MIS-C on in ammatory disorders. DOI: https://doi.org/10.1542/peds.2020-027763 Accepted for publication Dec 14, 2020 Address correspondence to Stacy A Kahn, MD, fl Coronavirus disease 2019 (COVID-19) is complaint of 3 weeks of hematochezia In ammatory Bowel Disease Center, Division of Gastroenterology, Hepatology and Nutrition, Boston increasingly associated with multisystem and worsening abdominal pain. He was Children’s Hospital, 300 Longwood Ave, Boston, MA inflammatorysyndromeinchildren(MIS- in his usual state of good health until 6 02115. E-mail: [email protected] C). MIS-C is a serious postinfectious weeks before, when he developed mild PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, syndrome resulting in cytokine storm rhinorrhea. The rhinorrhea resolved, 1098-4275). ∼ and multiorgan dysfunction often and 2 weeks later, he developed Copyright © 2021 by the American Academy of affecting the heart, the hematopoietic intermittent abdominal discomfort and Pediatrics system, and the gastrointestinal (GI) diarrhea, which progressed to 6 bloody FINANCIAL DISCLOSURE: The authors have indicated 1 tract. Here, we describe a novel and stools per day with severe generalized they have no financial relationships relevant to this timely case of an adolescent boy with an abdominal pain. He denied fevers, oral article to disclose. initial presentation of severe ulcers, myalgia, arthralgia, respiratory FUNDING: Dr Zhang is supported by the National inflammatory bowel disease (IBD) and symptoms, rash or headaches. Family Institutes of Health (T32 DK007477). Funded by the concurrent MIS-C. history was remarkable for a father National Institutes of Health (NIH). diagnosed with colonic cancer at age POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest CASE 43, currently in remission. There was no history of travel, trauma, or sick to disclose. A 16-year-old African American boy of contacts. Family history was negative Cape Verdean descent with no for autoimmune diseases or COVID-19. To cite: Sweeny KF, Zhang YJ, Crume B, et al. significant past medical history Inflammatory Bowel Disease Presenting With fl presented to an outside hospital On physical examination, the patient Concurrent COVID-19 Multisystem In ammatory Syndrome. Pediatrics. 2021;147(4):e2020027763 emergency department with a chief was thin, ill appearing, and met clinical Downloaded from www.aappublications.org/news by guest on September 30, 2021 PEDIATRICS Volume 147, number 4, April 2021:e2020027763 CASE REPORT criteria for systemic inflammatory ferritin level at 1116.0 ng/mL (range heterozygous ECMI and STAT3 single- response syndrome. He had a fever 10.0–320.0 ng/mL), a significantly nucleotide polymorphisms. with a maximum temperature of 38.0° elevated D-dimer level of 9.14 mg/mL C and remained febrile on the first day or fibrinogen-equivalent units (FEU) Immune workup revealed an elevated of hospitalization; he had a heart rate (range #0.50 mg/mL),andanelevated interleukin 2 receptor level at 1550 , of 165 beats per minute, a blood procalcitonin level of 0.65 ng/mL pg/mL (normal low level of 1033 pressure of 110/60 mm Hg, (range #0.09 ng/mL). The patient was pg/mL) and interleukin 6 level at 8 pg/mL (normal range ,5 pg/mL). a respiratory rate of 22, and an oxygen admitted to the gastroenterology Subsequently, SARS-CoV-2 saturation of 98% on room air. He service for further evaluation and immunoglobulin G testing results received 3 L of normal saline boluses treatment. Infectious diseases, returned positive, with a titer of 12 but remained tachycardic with a heart rheumatology, and immunology were (upper limit of normal 9), with repeat rate of ∼120 beats per minute. He had consulted. He received vitamin K and testing resulting in a titer of 16 and severe diffuse abdominal tenderness was started on 40 mg of intravenous a negative immunoglobulin M result. to palpation with involuntary guarding methylprednisolone daily. but no rebound or focal tenderness Because of concern for MIS-C, an and no hepatosplenomegaly. He had The patient was stabilized, and on electrocardiogram, an no cough, shortness of breath, rashes, hospital day 5, he underwent echocardiogram, troponin testing, and lymphadenopathy, icterus, esophagogastroduodenoscopy and N-terminal-pro-hormone brain conjunctivitis, joint tenderness or colonoscopy with biopsies. natriuretic peptide testing were swelling, and a normal perianal Esophagogastroduodenoscopy revealed performed, and results were all examination. severe gastritis and duodenitis. within normal limits. Colonoscopy revealed patchy moderate On hospital day 7, the patient had acute Laboratory evaluation was significant to severe colitis with a normal- worsening, with increasing large- for leukocytosis to 21 660 cells/mL appearing terminal ileum (Fig 1A). volume bloody stools, and the (range 5240–9740 cells/mL), Pathology from the upper GI tract methylprednisolone was increased to neutrophilia with an absolute revealed severe, chronic active 60 mg intravenous daily. Exclusive neutrophil count of 20 530 cells/mL duodenitis as well as chronic active enteral nutrition (EEN) was initiated. (range 2730–66 680 cells/mL) and gastritis. Notably, in the duodenum, On hospital day 10, because of severe lymphopenia with an absolute fl fi there was an in ammatory in ltrate ongoing symptoms unresponsive to lymphocyte count of 560 cells/mL focally involving medium-sized blood parenteral corticosteroids and EEN in – m (range 1030 2180 cells/ L). His was vessels. Although the computed the setting of MIS-C, he was given 1 mildly anemic, with a hemoglobin level tomography revealed mild ileitis, the g/kg of intravenous immunoglobin – of 10.4 g/dL, (range 11.0 14.3 g/dL) terminal ileum histopathology was (IVIg). Within 24 hours, his serum and a mean corpuscle volume of 83.4 normal. There was moderately to inflammatory markers and coagulation – fL (range 80.8 86.6 fL). His C-reactive severely active colitis in the ascending, laboratories began to downtrend (Fig protein (CRP) level was 22.14 mg/dL transverse, descending, and sigmoid 1C). Two days after receiving IVIg, he , (range 0.50 mg/dL) and his colon and mildly active colitis in the developed chest pain,afeverof40.0°C, erythrocyte sedimentation rate was 21 rectum (Fig 1B). Immunostaining and hypotension (blood pressure of – Helicobacter mm/hour (range 0 30 mm/hour). His results for cytomegalovirus, 95/36 mm Hg) and was transferred to albumin was normal at 4.0 g/dL. He (stomach), and SARS-CoV-2 were theICU.Hereceivedapackedred was mildly coagulopathic with an negative. Features of chronicity were blood cell transfusion for a hemoglobin international normalized ratio (INR) of seen in the duodenum, antrum and level that had dropped from 11.5 to 8.8 1.45 seconds. An abdominal computed corpus, and in several colon biopsies; g/dL and additional crystalloid fi tomography scan revealed trace free granulomata were not identi ed. The resuscitation. Within 24 hours, he had fl fi uid, mild ileitis, and signi cant diagnostic possibilities included IBD, stabilized and was transferred back to ascending and descending colitis with MIS-C, infection, or a combination the gastroenterology service. relative sparing of the transverse and thereof. The Prometheus IBD sgi sigmoid colon and rectum. Results of Diagnostic test was sent, and results The patient’sseruminflammatory infectious studies, including stool were consistent with Crohn disease, markers improved significantly on cultures, Clostridium difficile testing, with serology revealing an elevated steroids and IVIg; His CRP level blood cultures,
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