Esophageal ulcer and multisystem inflammatory syndrome after COVID-19: A case report

Abstract

BACKGROUND

Multisystem inflammatory syndrome in adults (MIS-A) is a rare but severe disease occurring several weeks after severe acute respiratory syndrome coronavirus 2 infection. It develops in adults with inflammation of different organs including the gastrointestinal tract, heart, kidneys, skin and hematopoietic system.

CASE SUMMARY

We present a 58-year-old Chinese man diagnosed with MIS-A. His chief complaints were fever, generalized fatigue and anorexia, accompanied with rashes on his back. Further examination showed cardiac, renal and liver injury. He had melena and gastroscopy indicated esophageal ulcer and severe esophagitis. Repeated blood and sputum culture did not show growth of bacteria or fungi. Antibiotic treatment was stopped due to unsatisfactory performance. His condition improved after prednisone and other supportive treatment.

CONCLUSION

Gastrointestinal involvement in MIS-A is not uncommon. Intestinal involvement predominates, and esophageal involvement is rarely reported. Esophageal ulcer with bleeding could also be a manifestation of MIS-A.

Key Words: Multisystem inflammatory syndrome; Esophageal ulcer; SARS-CoV-2; Gastrointestinal bleeding; Case report

Core Tip: Multisystem inflammatory syndrome in adults (MIS-A) is a rare but severe disease occurring several weeks after severe acute respiratory syndrome coronavirus 2 infection. We present a 58-year-old Chinese man diagnosed with MIS-A who had multiple organ damage. The patient had melena and gastroscopy indicated esophageal ulcer and severe esophagitis. His condition improved after prednisone and other supportive treatment. Clinicians should take MIS-A into consideration when they face unexplained inflammation and multiorgan damage. Gastrointestinal involvement in MIS-A is not uncommon. Intestinal involvement predominates, and esophageal involvement is rarely reported.

INTRODUCTION

Multisystem inflammatory syndrome (MIS) is a rare but severe disease related to COVID-19. It develops in adults and children with inflammation of different organs including the gastrointestinal tract, heart, kidneys, skin and hematopoietic system. When this phenomenon occurs in people aged > 21 years, it is called MIS in adults (MIS-A)[1]. Here, we report a case of MIS-A in a 58-year-old man after the 2022 severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) omicron variant in Beijing, China.

CASE PRESENTATION

Chief complaints

A 58-year-old male patient was admitted to hospital in January 2023, with a chief complaint of fever for 1 day, accompanied by fatigue, anorexia, mild cough, and whitish yellow sputum after becoming overtired.

History of present illness

The patient's highest body temperature was 39.6℃. He was first treated with empirical antibiotics of biapenem and supportive treatment. On day 6, melena suddenly occurred and his hemoglobin dropped to 64 g/L. Gastroscopy indicated a bleeding esophageal ulcer (Figure 1) and severe esophagitis. Gastrointestinal hemorrhage stopped after endoscopic hemostasis, abrosia, and proton pump inhibitor treatment. Body temperature dropped to 37°C after taking antipyretics, and rose to 39°C. His white blood cell count, serum creatinine, liver transaminase and total bilirubin continued to increase. The patient once received clindamycin infusion in a small clinic on day 1 from onset before hospitalization. However, allergy to clindamycin could not explain continuous deterioration of liver and kidney function after withdrawal. Empirical antibiotics changed to cefoperazone/sulbactam. Antibiotic treatment was stopped due to unsatisfactory performance while the patient’s body temperature began to drop to around 37°C. Cardiac, renal and liver insufficiency began to recover (Figure 2). The patient also regained his strength and appetite.

Figure 1 Computed tomography and endoscopy. A-I: Chest computed tomography (CT) on day 1 after onset of complained symptoms showing pulmonary inflammation; A-II: Chest CT on day 12 showing pulmonary edema; B-I: Endoscopic findings on day 9 showing esophageal ulcer 34 cm from the incisor; B-II: Endoscopic findings on day 19 showing smooth esophageal mucosa.

Figure 2 Changes of total bilirubin, creatinine and brain natriuretic peptide. TBIL: Total bilirubin; Cr: Creatinine; BNP: Brain natriuretic peptide.

History of past illness

He was diagnosed with SARS-CoV-2 infection by antigen test 5 weeks before, with symptoms of mild upper respiratory tract infection. The antigen test was negative 4 weeks before onset of fever.

Personal and family history

The patient had no specific personal history. His uncle was diagnosed with rhabdomyolysis and the cause was unknown.

Physical examination

On the first day of hospitalization, the patient had rashes on his back (Figure 3A). He had no palpable lymph node enlargement. Then his skin and sclera were gradually stained yellow. On day 12 of complained symptoms, the rash remained and turned darker (Figure 3B).

Figure 3 Patients with back rash at different times. A: Rash on day 2 of complained symptoms; B: Rash on day 12 of the symptoms.

Laboratory examinations

The polymerase chain reaction test for SARS-CoV-2 was negative. Immunoglobulin G antibody for SARS-CoV-2 was positive. His laboratory indexes showed abnormal liver function, hypoproteinemia, coagulation disorder, and acute renal insufficiency. On day 16, brain natriuretic peptide increased significantly. Repeated blood culture and sputum culture did not show growth of bacteria or fungi. Epstein–Barr virus DNA in the blood was positive, with low viral load. Blood analysis did not show atypic lymphocytes or elevated lymphocyte count. His autoantibody test and bone marrow examination were normal. No hemophagocytes were seen in the bone marrow.

Imaging examinations

Chest computed tomography (CT) revealed lung inflammation without typical features of viral pneumonia (Figure 1A). His abdominal imaging was normal without hepatosplenomegaly. CT on day 12 revealed pneumonedema (Figure 1A). Echocardiography showed reduced left ventricular systolic and diastolic function, as well as mild pericardial effusion. Venous thrombosis was found in the left lower extremity by ultrasound examination.

FINAL DIAGNOSIS

MIS-A.

TREATMENT

The patient was administered oral glucocorticoid 40 mg/d (approximately 0.5 mg/kg/d).

OUTCOME AND FOLLOW-UP

Ferrit and interleukin-6 were lower than before. A second gastroscopy on day 19 showed that the esophageal ulcer was completely healed (Figure 1B). The patient was discharged on day 32.

DISCUSSION

It was found that the rate of MIS-A was 9.9% and MIS-A-related mortality was 35.3%[2]. MIS usually occurred about 4 weeks after COVID-19 infection. In a recent systematic review[3], the organ systems most affected by MIS-A were hematological (92%), cardiovascular (87%), gastrointestinal (83%) and respiratory (74%). Our patient had multiple organ damage, including gastrointestinal tract, liver, heart and kidneys, at approximately 5 weeks after SARS-CoV-2 infection onset. Compared with patients with multisystem inflammatory syndrome in children (MIS-C), patients with MIS-A were more likely to have cardiac dysfunction and thrombosis[3,4]. MIS-C patients were more likely to present with dermatological and mucocutaneous manifestations[5].

In another systematic review, 73.4% of MIS-A patients had gastrointestinal symptoms[6]. Two cases of MIS-C undergoing endoscopy have been reported[7,8]. One had severe active gastroduodenitis and patchy colitis, and the other had complicated terminal ileitis, requiring ileocecal resection. Nonaka and colleagues[9] reported a 68-year-old male MIS-A patient whose intestine had extensive erosions from the duodenum to the ileum. The patient underwent arterial embolization and then small intestine resection because of hemorrhage. They also reported a 63-year-old man diagnosed with MIS-A with extensive erosion of the intestinal tract. A German medical team[10] reported a 27-year-old man diagnosed with MIS-A who had ileitis terminalis and colitis. Inflammation in the ileum and colon predominates in the gastrointestinal manifestations of MIS. However, upper gastrointestinal tract lesions demonstrated by imaging or endoscopic examination of MIS-A patients are rare, especially esophageal involvement. Here, we report a patient who had fever as the first symptom and subsequently developed esophageal ulcer. This case indicates that esophageal ulcer with bleeding could also be a manifestation of MIS-A. Timely endoscopic examination is helpful for diagnosis and treatment.

SARS-CoV-2 is implicated in the mechanism of MIS. The angiotensin-converting enzyme 2 receptor, which is used by SARS-CoV-2 to gain cell entry, is abundant in most organs[11]. The cytokine storm is also a potential driver of symptoms. Multiple cytokines including interleukin (IL)-1β, IL-6, IL-8, IL-10, IL-17, and interferon-γ are elevated in MIS-C patients[12]. MIS is also associated with thrombotic microangiopathy, which is characterized by microangiopathic hemolytic anemia and thrombocytopenia[13]. Future studies should elucidate the multi-factorial pathogenesis of MIS-A in order to identify the markers for treatment and prevention.

CONCLUSION

Gastrointestinal involvement in MIS-A is not uncommon. Intestinal involvement predominates, and esophageal involvement is rarely reported. We report a case of MIS-A with fever, rash and multiple organ dysfunction accompanied by esophageal ulcer. After glucocorticoid therapy, fever and multiple organ dysfunction were relieved, and the esophageal ulcer healed. The patient's prognosis was good.