Abstract Background Achalasia is an esophageal motility disorder resulting in chronic esophageal obstruction and stasis. Rapidly growing mycobacterium (RGM) pulmonary infections have been previously observed in individuals with underlying gastro-esophageal motility disorders. To date, pulmonary Mycobacterium wolinskyi infection (MWI) in the setting of achalasia has not previously been reported. Aims Our aim is to present a novel case of M. wolinskyi pneumonia in a patient with achalasia. We also searched the medical literature to identify previously reported associations between achalasia, chronic regurgitation symptoms and MWI. M. wolinskyi is an emerging clinical concern among RGMs. While primarily associated with post-operative, prosthetic joint and skin and soft tissue infections, M. wolinskyi pulmonary infections have not been documented to date in an immunocompetent patient. Methods We present a retrospective clinical case description of our case. We carried out a literature review from 1999 to Oct 2023, with the keywords "M. wolinskyi and infection. Results A 34-year-old male presented with a several-month history of productive cough, exertional dyspnea, fatigue, and unintentional weight loss of 17 kg over the past 6 months. Despite multiple courses of oral and intravenous antibiotics, there was no clinical improvement. His medical history included long-standing achalasia requiring previous pneumatic dilations. On admission, his chest X-ray revealed bilateral interstitial and alveolar infiltrates with consolidation. Routine microbiology and viral panels were negative. CT scan showed widespread bilateral consolidation and a dilated and fluid-filled esophagus. A timed barium swallow demonstrated dilated esophagus with irregular tertiary contractions and limited GE-J opening. Sputum cultures were positive for MWI on day 8 of admission (BACTEC MGIT). This was confirmed with lung biopsy demonstrating MWI with necrotizing granulomatous inflammation. The patient was prescribed a 12-month course of doxycycline, trimethoprim-sulfamethoxazole, and ciprofloxacin. Inpatient pneumatic dilation to 35 mm was performed. At discharge, his swallowing was subjectively improved, and a follow-up swallowing study noted improvement with no evidence of regurgitation. Conclusions Chronic esophageal obstruction provides an ideal lipid-rich environment for RGM infections. Related mycobacterial species have been isolated in pulmonary infections have been linked to chronic esophageal obstruction. To date, no cases of M. wolinskyi pulmonary infection with achalasia have been reported. We present a unique case of M. wolinskyi pulmonary infection in an immunocompetent patient with chronic achalasia, adding to our understanding of this emerging clinical concern. Funding Agencies None