Esophagitis Dissecans Superficialis Due to Possible Fishbone Ingestion: A Case Report

Abstract

Introduction:

Esophagitis Dissecans Superficialis (EDS) is a rare, underrecognized mucosal disorder of the esophagus. It is often associated with certain medications, systemic diseases, or direct esophageal trauma. Diagnosis is challenging due to its overlap with other esophageal conditions and the lack of standardized criteria.

Case presentation:

We report the case of a 38-year-old woman presenting with acute chest pain, dysphagia, and hematemesis following suspected fishbone ingestion. Initial endoscopic findings revealed sloughed esophageal mucosa with friability predominantly in the upper and mid-esophagus. Supportive management with high-dose proton pump inhibitor therapy and dietary modification resulted in rapid clinical improvement. Follow-up endoscopy at day 56 demonstrated complete resolution of mucosal abnormalities, and esophageal biopsies showed normal squamous epithelium. Based on the characteristic endoscopic findings, exclusion of alternative diagnoses, and benign, self-limited course, the presentation was most consistent with EDS, with fishbone ingestion considered a probable precipitating factor.

Conclusion:

This case highlights mechanical esophageal trauma due to possible fishbone ingestion as a rare but plausible trigger for EDS and underscores the importance of recognizing this entity in patients presenting with acute chest pain and upper gastrointestinal bleeding. Increased awareness of EDS may facilitate accurate diagnosis and prevent unnecessary interventions.

Introduction

Esophagitis Dissecans Superficialis (EDS) is a rare mucosal disease of the esophagus that is typically associated with certain medications use, infections, autoimmune mucosal disorders, or direct trauma.^1-5 EDS is likely underrecognized due to lack of diagnostic criteria and mimicking several other esophageal mucosal disorders. ⁶ It occurs more frequently with older age with a slight female predominance.^2,7 Symptoms include dysphagia, regurgitation, upper gastrointestinal bleeding, epigastric pain, heartburn, and atypical chest pain. ² Endoscopically, the sloughed esophageal linings appear pearly white with longitudinal fissures and circumferential cracks. ⁴ Histologically, EDS is characterized by desquamation and exfoliation of the superficial squamous epithelium, occasionally accompanied by bullous separation of the epithelial layers, parakeratosis, and variable levels of acute or chronic inflammatory changes. ⁶

The pathogenesis of the disease remains unclear. ⁷ However, various systemic diseases have been associated with EDS development, including bullous systemic lupus erythematosus, ⁸ pemphigus vulgaris, ³ celiac disease, ⁴ chronic kidney disease, ⁹ and HIV infection. ² Additionally, certain medications are known to induce EDS through esophageal irritation and localized esophageal injury. These include bisphosphonates (eg, alendronate), ¹⁰ nonsteroidal anti-inflammatory drugs (NSAIDs), ¹¹ antibiotics (eg, clindamycin), ¹² methotrexate, ¹³ and iron supplements ¹ among others.

Heavy smoking has also been identified as a potential etiological factor. ⁶ Furthermore, chemical irritants and thermal injuries, such as the ingestion of hot beverages and spicy foods, have been implicated. ¹⁴ Physical trauma, including nasogastric intubation, Mallory-Weiss tears, esophageal variceal sclerotherapy, and band ligation, has also been reported to contribute to the development of EDS. ⁵ Herein, we present a case of EDS following fishbone ingestion, which, to our knowledge, is a rare occurrence in literature. This case represents only the second reported instance of EDS associated with fishbone ingestion.

Case Summary

A 38-year-old Filipina woman presented to the emergency department with acute onset severe chest pain associated with vomiting of fresh blood and clots. Pain and vomiting started after having her lunch which contained fried fish, rice, and shrimp. She had a persistent sore throat and odynophagia, and she was not able to swallow her saliva. The patient denied the prior history of heartburn, chronic abdominal pain or dysphagia to food. No history of melena, bleeding per rectum, dizziness, or loss of consciousness. She did not suffer from any chronic medical illness, and she was not using any regular medications or analgesics. Systematic review was only significant for heavy menstrual periods, and she did not have previous surgeries or hospital admissions. Family history was unremarkable, and the patient did not smoke cigarettes or drink alcohol.

On examination, the patient was pale and anxious, but her vital signs were within the normal range. Cardiovascular, respiratory and abdominal examinations were normal. Digital rectal examination did not reveal melena or blood.

Laboratory investigations showed microcytic hypochromic anemia with a hemoglobin level of 6.7 g/dL (reference 12-15), ferritin of 2.5 µg/L (12-160), and low iron saturation 3.4% (15%-45%). Other laboratory tests including renal function test, liver function test, vitamin B12 level, and celiac serology were all within normal range. There were no previous laboratory investigations for comparison.

Imaging studies included plain chest X-ray, soft tissue neck X-ray and a contrast-enhanced computed tomography (CT) scan of the chest which did not reveal any evidence of retained foreign body, pneumomediastinum, or esophageal perforation. Given the fact that many fishbones are radiolucent and may not be visualized on plain radiography or CT images especially when small or oriented parallel to the imaging plane ¹⁵ and considering the temporal association with fish intake and persistent odynophagia, a clinically occult fishbone was still considered a plausible trigger.

The patient was maintained nil per os (NPO) and managed with intravenous fluids, along with intravenous esomeprazole 40 mg administered twice daily and ondansetron 4 mg by infusion every 8 hours for control of vomiting. An esophagogastroduodenoscopy (OGD) performed day 2 of admission which revealed severely sloughed esophageal mucosa with friability extending from the upper esophagus. The procedure was prematurely terminated due to the risk of perforation, and biopsies were not obtained. Supportive management was continued, and the patient was transitioned to a clear fluid diet. On day 5 of hospitalization, a repeat OGD showed linear sloughing of the esophageal mucosa, predominantly affecting the upper and mid esophagus, with spontaneous bleeding from the exposed mucosa. The lower esophageal mucosa appeared normal, with no evidence of stricture or mass lesions (shown in Figure 1).

Figure 1.

Antegrade esophagogastroduodenoscopy performed on day 5 demonstrating findings consistent with esophagitis dissecans superficialis. Linear mucosal sloughing with areas of spontaneous bleeding is indicated by arrows. The lesions predominantly involve the proximal esophagus ((A and B) forward view) and mid esophagus ((C and D) forward view). The distal esophagus ((E) forward view) and gastroesophageal junction ((F) forward view) appear intact without mucosal sloughing.

The patient demonstrated progressive clinical improvement and was able to tolerate a soft diet with only minimal odynophagia and chest discomfort. She was discharged after a 10-day hospitalization on oral pantoprazole 40 mg twice daily, with a scheduled follow-up in the gastroenterology clinic. Additionally, she received 1 g of intravenous ferric carboxymaltose for iron repletion and was referred to gynecology for evaluation of heavy menstrual bleeding.

At a follow-up on day 35, the patient was completely asymptomatic. A repeat OGD on day 56 revealed complete resolution of the esophageal mucosal changes (shown in Figure 2). A total of 4 biopsies were taken, 2 from the upper and 2 more from the mid-esophagus. Histopathological examination using routine hematoxylin and eosin stain showed normal stratified squamous epithelium with no evidence fir inflammation, dysplasia, fungal organisms, or viral cytopathic changes which confirmed complete mucosal regeneration. Repeated full count at week eight showed resolution of anemia with a hemoglobin of 12 mg/dL. However, she continued to follow-up with gynecology for menorrhagia due to uterine fibroids.

Figure 2.

Follow-up esophagogastroduodenoscopy on day 56 showing complete resolution of the previously observed mucosal sloughing and bleeding, with restoration of normal esophageal mucosa.

Discussion

We report a rare case of EDS in a middle-aged woman that is likely related to fishbone ingestion. Unlike most reported cases, this patient was younger and did not exhibit traditional risk factors for EDS. Symptoms developed abruptly following food ingestion, with esophageal involvement primarily localized to the upper and middle portions of the esophagus. Despite the initial severity of symptoms, the patient achieved complete recovery, and follow-up endoscopy demonstrated full resolution of the previously observed mucosal changes.

To the best of our knowledge, this is the second reported case of fishbone ingestion-related EDS. Karagozian and Gan described EDS involving the entire esophagus in a 28-year-old pregnant woman after eating fish that contained numerous small bone particles followed by vinegar ingestion in attempt to soften the bone as a rescue remedy by the patient. OGD showed completely denuded mucosal linings with tethering at the gastroesophageal junction with clotted blood in the stomach wrapped in a 15 cm gray membranous tissue that was composed of normal squamous cells. The patient achieved complete recovery without residual inflammation or stricture. Although fish bone might have triggered mucosal injury, EDS was likely propagated by acid ingestion and pregnancy related esophageal motility impairment. ¹⁶ In comparison, the patient described in our case did not have risk factors for esophageal dysmotility and was not exposed to additional mucosal irritative material.

Although a fish bone was not visualized on the imaging modalities in this case, previous comparative studies have demonstrated a low sensitivity for plain radiography (approximately 39%) compared with CT scans (around 90%) in identifying fish bones. Many fish bones may also appear radiolucent on imaging, and both plain radiography and CT have imperfect sensitivity for detecting thin or partially embedded bones, particularly depending on their size, species, and orientation; therefore, clinical suspicion may persist despite negative imaging findings. ¹⁵ Therefore, the absence of imaging evidence for an esophageal fish bone does not exclude the possibility of fish bone related EDS in this case.

Another notable observation in this case is the profound iron deficiency anemia at presentation. Although the patient presented with acute odynophagia and hematemesis, she remained hemodynamically stable, and digital rectal examination did not reveal melena. The markedly low hemoglobin and ferritin levels were therefore considered most consistent with chronic iron-deficiency anemia related to heavy menstrual bleeding, with possible minor contribution from the episode of hematemesis. Stool testing for fecal occult blood was not performed during hospitalization. However, there was no clinical evidence of ongoing gastrointestinal bleeding during follow-up. The patient received intravenous iron replacement and was referred for outpatient follow-up with both internal medicine and gynecology clinics, where we noticed significant improvement in the hemoglobin levels.

The pathophysiology of EDS is thought to involve superficial epithelial injury leading to necrosis and subsequent sloughing of the squamous mucosa in large sheets. ⁷ In this case, a sharp foreign body such as a fishbone may have induced focal linear epithelial trauma, initiating superficial mucosal necrosis without deeper tissue involvement. Esophageal peristalsis and luminal shear stress may then propagate epithelial cleavage along the squamous layers, resulting in characteristic longitudinal sloughing. Subepithelial capillary disruption likely accounts for the observed superficial bleeding, while preservation of the basal epithelial layer allows for rapid and complete mucosal regeneration. Acid exposure may further exacerbate epithelial separation in the absence of protective mucosal integrity.^6,7,16 Importantly, the absence of known systemic, medication-related, or autoimmune risk factors supports isolated mechanical trauma as a sufficient trigger in this patient

Notably, in the 2 cases of fish bone-related EDS, the patients age was younger than the median age of around 70 described in most of the previous studies.^6,7,14 Both cases affected women which was consistent with the slight female predominance observed in earlier 2 case series of 41 and 7 cases respectively.^7,14 Whereas another case series of 12 patients showed higher prevalence in males. However, given the small number of reported cases it would be difficult to draw definite conclusions on the effect of sex on disease prevalence. The older age of other reported cases is likely related to the underlaying comorbidities increasing the risk for EDS including infections, chronic kidney disease and mucosal disorders such as pemphigus. Another distinctive feature of this case is the predominant involvement of the proximal esophagus in contrast to the involvement of middle and lower parts of the esophagus in most other cases.^6,7,14

In this case, the patient presented acutely with chest pain, dysphagia, and hematemesis. This presentation contrasts with the typical diagnostic context of EDS, which is often identified during endoscopy performed for gradually onset symptoms with less severe manifestations. In a cohort of 41 patients treated at a tertiary care center, the diagnosis of EDS was predominantly incidental, reflecting limited clinical recognition of its characteristic endoscopic features. ⁷ The underreporting and underdiagnosis of EDS may be partly attributed to the infrequent occurrence of the condition and the frequent contamination of esophageal specimens with bacterial or fungal organisms. ¹⁷ Currently, there is no universally accepted diagnostic criterion for EDS; however, Hart et al ⁷ proposed that diagnosis should require meeting 3 endoscopic criteria: sloughed strips of esophageal mucosa measuring at least 2 cm in length, a normal appearance of the underlying esophageal mucosa, and the absence of ulcerations or friability in the immediately adjacent mucosa. In the present case, repeat endoscopy demonstrated longitudinal sheets of sloughed mucosa extending through the upper and mid esophagus, with preservation of the deeper mucosal architecture and complete resolution on follow-up endoscopy. While mucosal friability was present acutely, this was confined to the exposed sloughing surface and resolved entirely, supporting a superficial and reversible epithelial process consistent with EDS rather than deep mucosal injury. Infectious etiologies were considered unlikely given the absence of immunosuppression, lack of endoscopic ulceration or exudates, and complete spontaneous resolution without antimicrobial therapy. Autoimmune bullous diseases were considered unlikely due to the absence of cutaneous or mucosal involvement elsewhere, and intramural esophageal dissection was ruled out by imaging and endoscopic appearance.

The primary approach to managing EDS involves addressing the underlying cause, when identifiable, along with the use of high-dose proton pump inhibitors, as employed in this case.^1-4,8-17 Despite these considerations, the clinical course of EDS generally appears to be benign, with frequent resolution of endoscopic findings.^7,17

Conclusion

This case highlights a rare and diagnostically challenging presentation of ESD most likely precipitated by mechanical trauma from fishbone ingestion. Notably, EDS continues to be a diagnosis of exclusion, particularly in acute presentations following esophageal trauma. It requires careful evaluation of infectious, necrotizing, autoimmune, and structural causes. Although the absence of acute-phase histology limits absolute diagnostic certainty, the characteristic endoscopic appearance, exclusion of alternative etiologies, benign clinical course, and complete endoscopic resolution strongly support the diagnosis of EDS while making infectious, caustic or necrotizing causes highly unlikely. Clinicians should consider EDS in patients presenting with acute chest pain and upper gastrointestinal bleeding following esophageal trauma, as early recognition and conservative management are associated with excellent outcomes.