Acute esophageal necrosis after cellulitis in an obese patient with diabetes mellitus.

A 59-year-old obese Japanese man with poorly controlled type 2 diabetes mellitus presented with severe heartburn for 3 days after inguinal cellulitis and exacerbated glycemic control, without any signs of upper gastrointestinal bleeding. The patient had a high plasma glucose level (34.0 mmol/L) and was dehydrated. Emergent esophagogastroduodenoscopy showed black discoloration predominantly affecting the lower esophagus; thus, acute esophageal necrosis (AEN) was diagnosed. This black discoloration was not present on esophagogastroduodenoscopy 20 days prior to presentation, and disappeared 6 days after conservative treatment. To conclude, acute esophageal necrosis should be considered if a patient in marked hyperglycemic status presents with unendurable heartburn, even when upper gastrointestinal bleeding is not observed or recent esophagogastroduodenoscopy was unremarkable.

Introduction

Exacerbated glycemic profiles after infectious disease in patients with diabetes mellitus are frequently encountered in clinical practice. Marked hyperglycemia sometimes dehydrates patients through osmotic diuresis, and gives rise to renal insufficiency. Acute esophageal necrosis (AEN) is a rare but potentially life‐threatening syndrome that preferentially affects dehydrated elderly men with hyperglycemia, and arises together with upper gastrointestinal bleeding in almost all such patients1. Herein, we report a case of AEN without any initial signs of upper gastrointestinal bleeding in whom subsequent esophagogastroduodenoscopy (EGD) led to a diagnosis of AEN.

Case Report

A 59‐year‐old Japanese man with type 2 diabetes mellitus presented with spreading erythema in the inguinal region, followed by a deteriorated glycemic profile for 1 week, and heartburn for 3 days. His diabetes mellitus was unsatisfactorily controlled: the glycated hemoglobin level was 9.0%, despite 2,000 mg/day metformin, 4–6 units of insulin glulisine before meals and 20 units of insulin glargine at bedtime. The severe heartburn prevented the patient from eating solid meals, although a small amount of a liquid meal with suspended metformin and insulin was taken, and an accelerated deterioration in glycemic control was subsequently noted. Episodes of melena, hematemesis or coffee‐ground emesis were not apparent.

The patient was obese (body mass index 33.6 kg/m2), normotensive but tachycardic, and had a low‐grade fever and dry mucous membranes. Right inguinal cellulitis with surface necrotic tissue complicated by a subcutaneous abscess (Figure 1) was observed. Laboratory analysis revealed a white blood cell count of 17,100/μL, blood urea nitrogen at 37.7 mg/dL, creatinine at 0.98 mg/dL, an estimated glomerular filtration rate of 61.5 mL/min/1.73 m2 (a drop from a baseline level of 84.9 mL/min/1.73 m2), C‐reactive protein at 25.4 mg/dL, plasma glucose at 34.0 mmol/L and a glycated hemoglobin level of 10.1%. Diabetic ketosis without acidosis was also observed.

Figure 1

Cellulitis with a subcutaneous abscess in the lower inguinal region.

Esophagogastroduodenoscopy during a medical checkup 20 days before presentation had shown only short‐segment Barrett's esophagus (Figure 2a), whereas emergent EGD on arrival showed diffuse erosive esophagitis with black discoloration predominantly affecting the lower esophagus and abruptly interrupted at the gastroesophageal junction (Figure 2b); thus, a diagnosis of AEN was made.

Figure 2

Esophagogastroduodenoscopy findings. (a) Twenty days before presentation. (b) On admission. (c) On hospitalization day 6.

The patient was managed by oral intake restriction, fluid replacement with isotonic solution, intravenous omeprazole 20 mg twice daily, intravenous insulin infusion, piperacillin–tazobactam 4.5 g three times daily, and by incision and drainage of the abscess. Prerenal azotemia, decreased estimated glomerular filtration rate and ketosis swiftly improved, with 8–10 mmol/L glucose achieved on hospitalization day 5. Follow‐up EGD on day 6 showed a marked improvement of AEN (Figure 2c). Thus, the intravenous omeprazole was switched to vonoprazan 20 mg orally once daily, and the intravenous insulin was switched to multiple daily injections in accordance with a restarted oral intake. The cellulitis healed by hospitalization day 16, and the patient was discharged.

Written informed consent was obtained from the patient for publication of this report. Formal ethics approval was waived, because this is a case report.

Discussion

AEN, a syndrome endoscopically characterized by a diffuse black discoloration affecting the distal esophagus, was first described by Goldenberg et al.2 in 1990. Although previous endoscopy series have described AEN as being very rare, with an estimated prevalence rate of between 0.01 and 0.28% of endoscopies, early diagnosis and management are required due to possible esophageal perforation and high mortality rates3. Indeed, the overall mortality of patients with AEN was found to be 31.8%4. AEN frequently occurs in elderly persons and has a clear male predominance; nearly 90% of cases manifest melena, hematemesis or coffee‐ground emesis1, 4. Hyperglycemia and diabetes mellitus are recognized risk factors for AEN because of a frequent co‐existence; AEN with diabetic ketoacidosis has also sometimes been reported1, 4, 5, 6, 7.

The most plausible explanations for the etiology of this case include gastroesophageal acid reflux, weakened mucosal defense and hypoperfusion, as these are associated with the developmental mechanisms of AEN4. Pre‐existing Barrett's esophagus indicates long‐standing gastroesophageal reflux. Critical illness (severe cellulitis, after marked hyperglycemia and renal insufficiency in the present case) leads to diminished acid buffering, resulting in a weakened esophageal defense3. Furthermore, hemodynamic instability related to severe infection and dehydration due to hyperglycemic osmotic diuresis, suspected based on a physical assessment and laboratory findings, can involve rapid injury to esophageal tissue3.

The pitfalls of AEN management are highlighted in the present case. First is the lack of any signs of upper gastrointestinal bleeding. Because 7% of cases are accompanied by the acute life‐threatening complication of esophageal perforation, which requires life‐saving surgical intervention, the early recognition and management of AEN is vital1, 4. However, laboratory findings of AEN are not specific and largely due to the underlying disease1, 4. Therefore, clinicians carry out EGD when patients with multiple risk factors of AEN complain of heartburn, even in the absence of signs of upper gastrointestinal bleeding. Second is the dramatic difference between EGD findings on admission and the latest, coincidentally obtained, premorbid EGD findings. The development of AEN is thought to be rapid, having occurred within 18 h in one case8. This present case highlights how AEN can develop when previously trivial endoscopic findings were yielded, thus suggesting that a recent EGD showing unremarkable findings cannot rule out AEN in clinical practice.

The deterioration in glycemic profile after preceding infectious disease in patients with poorly controlled diabetes mellitus is frequently encountered in a clinical setup. Such patients are sometimes dehydrated and thus at high risk of AEN. Clinicians should consider AEN and carry out timely EGD if a diabetes patient with a severely elevated glucose level complains of unendurable heartburn, even when signs of upper gastrointestinal bleeding are absent or a recent EGD was unremarkable.

Disclosure

The authors declare no conflict of interest.