Case Report
 
Esophageal Actinomycosis, A Rare Complication After Self-Expanding Metallic Stent Deployment
 
Chandrasekar Thoguluva Seshadri1, Sathiamoorthy Suriyanarayanan1, Gokul Bollu Janakan1, Raja Yogesh Kalamegam1, Prasad Menta Sanjeevaraya2, Viveksandeep Thoguluva Chandrasekar1
Department of 1Gastroenterology and 2Surgery,  Med India Hospitals, Chennai.



Corresponding Author
:
Dr Chandrasekar Thoguluva Seshadri 
Email: tscmedindia@yahoo.com 


Abstract

Actinomycosis is a rare, sub-acute to chronic bacterial infection caused by Actinomyces species which are facultative, anaerobic, gram positive bacilli. It is most frequently caused by species Actinomyces israelii. They are mainly present as saprophytes in the soil. In humans, they can occur as commensals of the oral cavity, gastrointestinal tract and vagina. Actinomycosis is preceded by some known insults to the native mucosa. It is characterized by contiguous spread, granulomatous inflammation and formation of multiple abscesses and sinus tracts that may discharge sulphur granules. Though Actinomyces infection can occur in abdomino-pelvic, pulmono-thoracic and cervico-facial areas, oesophageal involvement is an extremely rare occurrence in immunocompetent persons. Here we report a rare case of oesophageal actinomycosis in a 50-year-old lady, following the deployment of a partially covered Self Expanding Metallic Stent (PCSEMS) elsewhere for an indeterminate mid oesophageal stricture. It is noteworthy that this is the first case following oesophageal metallic stenting, reported from India. 

Case Report

A 50 year old lady presented with a history of progressive dysphagia to solids and liquids for the past 3 months. Four months ago, she was diagnosed to have an oesophageal stricture at 25 cm, for which a PCSEMS was deployed elsewhere as it was considered malignant. 
On physical examination, she was thin built and malnourished. Her blood investigations showed low haemoglobin of 85 g/L and serum albumin of 28 g/L. The oesophagoscopy revealed a proliferative circumferential tissue overgrowth at 20 cm at the proximal end of PCSEMS narrowing the lumen not admitting the endoscope further (Figure 1). Biopsy was taken from the tissue in-growth. 



       Computed Tomography of the chest revealed an oesophageal metallic stent in the mid and lower third of oesophagus with tissue in-growth into the proximal end of the stent and wall thickening in the mid third of the oesophagus (Figure 2A & 2B). The biopsy and histological examination showed discrete sulphur granules within the granulation tissue in the ulcerated oesophageal tissue consistent with infection by Actinomyces species (Figure 3A)
This patient was treated with a course of intravenous penicillin-G for a month. She had good relief of chest pain and odynophagia with partial alleviation of dysphagia. She was reassessed with upper GI scopy which revealed partial regression of hyperplastic tissue overgrowth. Repeat biopsy from the hyperplastic tissue revealed only granulation tissue without any evidence of actinomycotic infection or malignancy (Figure 3B). This persistent hyperplastic tissue was subsequently ablated by Argon plasma coagulation. 




Discussion

Actinomyces are facultative anaerobic, gram-positive bacilli that colonize oral and GI tract as well as the vaginal mucosa.1 Although these organisms have a low degree of pathogenicity, mucosal disruption can result in chronic granulomatous and suppurative infection that can involve almost any organ system.2 Over time, the bacteria can cross natural anatomic boundaries, resulting in multiple abscesses, draining sinus tracts, and ?stula formation.3
Esophageal actinomycosis has been described in both immunocompetent and immunocompromised patients. Dysphagia and odynophagia are the most common presenting symptoms.4 Endoscopy may reveal multiple discrete round ulcers. The primary diagnostic tools remain Gram stain and histology which may show typical microscopic findings that include necrosis with yellowish sulphur granules and filamentous Gram-positive fungus-like pathogens.2 De?nitive diagnosis with positive cultures requires 5 to 7 days but is seen in only a minority of cases.5 Barium oesophagogram may demonstrate deep ulcers with fistulas and multiple sinus tracts. CT may demonstrate a thickened oesophageal wall in early stages with or without sinus tracts.  
The  thoracic and abdominal forms of actino-mycosis is treated with a 4-6 week course of intravenous penicillin - G, 3-4 million units every four hours, followed by oral pencillin or amoxicillin for 6-12 months. Prolonged (6 to 12 months) high doses of penicillin G or amoxicillin is needed to facilitate the drug penetration in abscesses and infected tissues. Tetracycline is the preferred antibiotic for individuals with penicillin allergy, although minocycline, erythromycin, clindamycin, ceftriaxone, and imipenem are available alternative treatments.

References
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