Recurrent idiopathic esophageal perforation complicated by thyroid abscess: a case report and review of the literature

Abstract

Background

Idiopathic esophageal perforation complicated by thyroid abscess formation constitutes a rare and severe clinical emergency. The precise pathogenesis of this condition remains elusive, and its potential for recurrence necessitates further investigation to establish evidence-based, personalized therapeutic protocols.

Case Description

We present a case of a 41-year-old female who initially presented with a spontaneous cervical esophageal perforation associated with an ipsilateral thyroid abscess. The patient underwent prompt surgical intervention consisting of meticulous debridement and drainage, facilitated by intraoperative neuromonitoring to preserve recurrent laryngeal nerve function and thyroid integrity. The postoperative course was uneventful, and the patient was discharged with preserved thyroid function. However, a recurrence manifested 10 months postoperatively. In light of the well-localized abscess and the patient’s hemodynamically stable condition, a conservative management strategy was instituted. This approach, comprising targeted antimicrobial therapy and vigilant clinical surveillance, resulted in complete resolution without the need for further surgical intervention.

Conclusions

This case provides substantive clinical insights for formulating individualized management strategies for idiopathic esophageal perforation. A review of the extant literature suggests that underlying anatomical anomalies, such as congenital weakness or diverticula, may predispose individuals to recurrent episodes. This underscores the imperative of tailoring treatment—ranging from aggressive surgical intervention to meticulous conservative management—based on a comprehensive assessment of infection severity, anatomical involvement, and the patient’s overall clinical status. Further multicentric studies are warranted to elucidate the etiopathogenesis and to refine risk-stratified treatment algorithms for this uncommon but potentially life-threatening condition.

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Key findings

• This case highlights recurrent idiopathic esophageal perforation complicated by a thyroid abscess, a rare and life-threatening emergency. Recurrence was associated with the formation of an esophageal diverticulum, suggesting a potential anatomical predisposition.

What is known and what is new?

• Idiopathic esophageal perforation is uncommon and often occurs without clear triggers, while thyroid abscesses are rare due to the gland’s protective features. Concurrent presentation significantly increases the risk of severe infection and mediastinitis.

• This case provides new insights into recurrence mechanisms, linking it to diverticulum formation post-perforation. It also demonstrates successful conservative management during recurrence, contrary to typical surgical approaches for complex infections.

What is the implication, and what should change now?

• This case underscores the need to investigate anatomical abnormalities, such as diverticula, in patients with recurrent esophageal perforation. High-resolution imaging and long-term surveillance should be prioritized to identify underlying structural causes. For localized infections without systemic compromise, conservative management may be considered to avoid unnecessary surgery.

Introduction

Esophageal perforation represents a life-threatening emergency that is often associated with delayed diagnosis due to its diverse and sometimes obscure etiologies (1). These etiologies include iatrogenic injury, spontaneous rupture, malignancy, benign ulcers, trauma, caustic ingestion, foreign body impaction (2), and rare idiopathic causes (3). Thyroid abscesses are rarely encountered in clinical practice due to the gland’s unique anatomical and physiological characteristics, making early diagnosis and management essential. When esophageal perforation occurs concurrently with a thyroid abscess, the risk of sepsis and mediastinitis is markedly increased, owing to the complex anatomical structure of the cervical fascial planes (4). This report describes a rare case of recurrent idiopathic cervical esophageal perforation initially complicated by a thyroid abscess, managed with surgical intervention. During recurrence, conservative treatment was administered, resulting in favorable outcomes. A literature review is included to explore potential etiologies, therapeutic strategies, and recurrence mechanisms, with the aim of informing clinical management in similar cases. We present this article in accordance with the CARE reporting checklist (available at https://acr.amegroups.com/article/view/10.21037/acr-20251-260/rc).

Case presentation

A 41-year-old female presented with a 1-week history of neck pain and swelling. She denied any history of vomiting, chest pain, or foreign body aspiration. Laboratory investigations revealed elevated inflammatory markers. Cervical computed tomography (CT) revealed an abscess extending from the anterior cervical space to the left anterior mediastinum (1.7 cm × 0.9 cm), with a low-density lesion in the left thyroid lobe (0.2 cm) (Figure 1). Esophagography revealed a perforation on the left lateral wall of the upper esophagus (Figure 2). A diagnosis of cervical esophageal perforation with a concomitant thyroid abscess was established. Urgent surgical drainage via the parapharyngeal space was conducted under intraoperative nerve monitoring (IONM). The esophageal perforation site was explicitly explored and dissected, located on the left lateral wall, measuring approximately 0.3 cm, with edematous but viable marginal tissue. After local debridement, a closed negative-pressure drainage tube was placed. The thyroid abscess was managed with simple drainage without partial thyroidectomy. Intraoperatively, a purulent collection was identified between the posterior aspect of the upper left thyroid lobe and the surface of the esophagus. Partial liquefaction of the thyroid tissue (2 cm × 1.5 cm) was noted. Postoperatively, a nasojejunal feeding tube was inserted, and intravenous antibiotics were administered. The patient was discharged on postoperative day 25, following confirmation of esophageal healing via contrast esophagography (Figure 3).

Figure 1.

Neck CT for the initial perforation. The red circle indicates the abscess in the left thyroid lobe and anterior mediastinum. CT, computed tomography.

Figure 2.

Esophagography for the initial perforation. The red circle indicates the site of contrast leakage at the left lateral wall of the upper esophagus.

Figure 3.

Esophagography was reviewed on postoperative day 25.

Ten months later, the patient presented again with throat discomfort and left-sided cervical pain. Cervical CT revealed a small abscess measuring 1.4 cm (Figure 4), and esophagography demonstrated a recurrent perforation in the left lateral wall of the upper esophagus (Figure 5). Conservative treatment, including intravenous antibiotics and nasogastric feeding, was initiated. Follow-up CT on day 4 revealed resolution of the abscess and the presence of an esophageal diverticulum (Figure 6). On day 10, contrast esophagography demonstrated complete closure of the perforation (Figure 7). Subsequent endoscopy confirmed the presence of the esophageal diverticulum (Figure 8). Following recovery, the patient was placed on a surveillance regimen due to the small (0.3 cm × 0.2 cm), asymptomatic diverticulum. This plan entailed periodic imaging and endoscopic follow-up, with surgical intervention reserved for potential future enlargement or symptom recurrence.

Figure 4.

Neck CT for the second perforation. The red circle indicates the recurrent small abscess adjacent to the esophagus. CT, computed tomography.

Figure 5.

Esophagography for the second perforation. The red circle indicates the recurrent contrast extravasation at the previous perforation site.

Figure 6.

Neck CT on day 4 of the second admission showed an esophageal diverticulum. The red circle indicates the newly formed esophageal diverticulum. CT, computed tomography.

Figure 7.

Ten days after the second admission, esophagography was repeated.

Figure 8.

Gastroscopy revealed an esophageal diverticulum.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration and its subsequent amendments. Written informed consent for publication of this case report and accompanying images was not obtained from the patient or the relatives after all possible attempts were made.

Discussion

Management of esophageal perforation must be individualized according to the perforation site, infection severity, and the patient’s systemic condition. In this case, the initial perforation was complicated by a thyroid abscess and mediastinal infection, constituting a severe and complex infectious process. Prompt surgical debridement was essential for source control and mortality reduction. The literature supports that surgical drainage in cases involving cervical or mediastinal abscesses can effectively prevent sepsis and multi-organ failure (2). In contrast, the second perforation presented with a localized abscess without systemic toxicity. Conservative management with antibiotics and enteral nutritional support was considered appropriate. Recent studies have demonstrated that non-surgical approaches may be safe and effective in selected cases (5). This case underscores the importance of context-specific treatment planning to minimize surgical trauma and preserve organ function.

Thyroid abscesses are rare clinical entities, largely due to the gland’s protective anatomical and physiological features, including rich vascularization, high iodine concentration, efficient lymphatic drainage, and a fibrous capsule that serves as a physical barrier against infection (6). The early symptoms are often nonspecific, such as neck swelling and pain, and imaging characteristics are typically non-distinctive, which frequently leads to delayed diagnosis (7). Surgical management options generally include either simple abscess drainage or drainage combined with partial thyroidectomy (1). In the present case, drainage alone was performed under IONM, allowing for maximal preservation of thyroid function. This approach was chosen based on two primary considerations: first, cervical infection can result in edema and dense adhesions between the thyroid and adjacent structures, such as the esophagus, obscuring anatomical landmarks and increasing the risk of recurrent laryngeal nerve injury during resection. IONM has been shown to significantly reduce the incidence of such nerve injuries by facilitating real-time identification of neural structures during surgery (8). Second, the abscess was localized to the posterior portion of the left thyroid lobe, and resection might have resulted in unnecessary tissue loss and potentially permanent hypothyroidism. In contrast, drainage combined with appropriate antibiotic therapy yielded favorable outcomes without compromising endocrine function (5). Nevertheless, in more complex cases, particularly those involving fistula formation or extensive tissue destruction, thyroidectomy may still be required to achieve definitive infection control (9).

The initial perforation was most likely idiopathic in nature, corresponding to the subtype described by Sohda et al., which distinguishes idiopathic from post-emetic forms of spontaneous esophageal rupture (3). The idiopathic type typically lacks a triggering event such as emesis and frequently occurs along the left lateral esophageal wall, a region considered anatomically susceptible due to its structural thinness (10). In this case, non-specific cervical symptoms, absence of vomiting, and lack of foreign body ingestion or trauma supported the diagnosis. Elevated C-reactive protein (CRP) levels were indicative of delayed recognition and active inflammation (11). However, the clinical features and pathogenesis of idiopathic esophageal perforation remain to be further confirmed through multicenter studies, standardized diagnostic procedures, and expanded sample sizes. In this case, given the relatively small perforation size, we did not perform suture repair but instead placed a drainage tube combined with a pressure dressing, aiming to promote granulation tissue growth and scar repair, thereby achieving secondary healing. Recurrence was likely related to anatomical abnormalities, including the development of an esophageal diverticulum (12). Although not observed on initial imaging, a diverticulum was later detected on follow-up, suggesting that it may have formed secondary to abnormal tissue remodeling and post-perforation fibrosis (13). These changes could lead to decreased tissue elasticity and localized bulging under intraluminal pressure during deglutition. Furthermore, the transition zone between fibrotic and healthy tissue may become structurally compromised, rendering the site more prone to secondary perforation. Infectious and inflammatory damage may further compromise esophageal wall integrity, contributing to focal weakness and diverticular formation. The diverticulum, likely a traction-type diverticulum resulting from periesophageal inflammation and fibrosis, arose through a combination of scarring, inflammation, and tissue necrosis (14). Despite its rarity, such structural alterations can significantly increase the risk of recurrence. Differential diagnosis must also include congenital malformations, esophageal motility disorders, or external compressive lesions such as neoplasms. Notably, invasive thyroid malignancies (e.g., thyroid angiosarcoma) eroding the esophageal wall may also precipitate perforation and abscess formation (15). Advanced diagnostic modalities, including high-resolution manometry and endoscopic ultrasound, are recommended to exclude these possibilities, and long-term surveillance remains essential to prevent further recurrence.

Conclusions

This case underscores the need for individualized treatment strategies in the management of idiopathic esophageal perforation. Surgical intervention under IONM during the initial episode enabled accurate debridement and optimal preservation of thyroid function. Upon recurrence, conservative management with intravenous antibiotics and enteral nutrition was sufficient to achieve perforation closure and effective infection control. This case suggests that treatment planning must take into account anatomical anomalies, immune status, and the extent of infection. In patients with recurrent perforation, clinicians must remain alert to the possibility of esophageal diverticula or occult infectious foci. The use of high-resolution imaging, combined with long-term follow-up, is recommended to further elucidate the underlying etiology and recurrence mechanisms. Optimizing treatment strategies through these approaches has the potential to reduce complications and enhance long-term outcomes.

Supplementary

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Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration and its subsequent amendments. Written informed consent for publication of this case report and accompanying images was not obtained from the patient or the relatives after all possible attempts were made.