TOPIC: Chest Infections TYPE: Medical Student/Resident Case Reports INTRODUCTION: Fibrothorax occurs as a long-term complication of untreated pleural infections. Well-known causes of fibrothorax include recurrent parapneumonic effusions, empyema, and frank hemorrhage in the pleural space. Here we present a case of massive fibrothorax secondary to silent aspiration pneumonias in a patient with Down's syndrome. CASE PRESENTATION: A 24-year-old male with Down's syndrome and autism presents with a complaint of right-sided back pain and fever for two days. The patient is non-verbal at baseline but is able to communicate with his mother, who is the primary caregiver. He has a fixed routine throughout the day, owing to his autism, and his mother takes excessive precautions in tracking his symptoms and keeping his healthcare appointments.In the ER, the patient was hypoxic and tachypneic; initially requiring non-invasive ventilation, and subsequently mechanical ventilation with admission to the ICU. Work-up including chest radiograph revealed complete opacification of the right hemithorax, while the CT chest revealed a large right-sided pleural effusion, consolidation, and a thick pleural peel. Broad-spectrum antibiotics were initiated, and the patient subsequently underwent a right-sided VATS and lung decortication. Intraoperative findings revealed massive fibrothorax without frank pus. Blood and respiratory cultures were negative, surgical culture from pleural peel, however, grew Streptococcus intermedius (S. intermedius). Pathology demonstrated granulation tissue with abundant adherent fibrin and fibrinopurulent exudate, compatible with empyema. On day 7, he was transferred out of the ICU after showing marked clinical improvement. DISCUSSION: Diffuse pleural thickening or fibrothorax occurs due to severe pleural space inflammation. The development of empyema is an uncommon complication, with a reported incidence of 2-3% of all pneumonias. We hypothesize the etiology in our patient to be attributed to recurrent silent aspirations in a patient with Downs syndrome. Anatomical and functional abnormalities including macroglossia, poor neuromotor control, and gastroesophageal reflux make these patients aspiration-prone, which quite often are silent. This is further supported by the presence of S. intermedius in the pleural culture. This bacterium is commonly found in the oral flora but has been associated with brain, liver and thoracic empyema. Recent literature review revealed a limited number of reported cases of pneumonia caused by S. intermedius. Decortication is an effective strategy for treating symptomatic patients, regardless of cause, when significant underlying parenchymal disease has been excluded, as was done in our patient. CONCLUSIONS: It is important for clinicians to be aware of uncommon causes of fibrothorax, such as seen in this case of recurrent silent aspirations, without recurrent pleural effusions, leading to strep intermedius-associated pleural fibrosis. REFERENCE #1: Hannoodi F, Ali I, Sabbagh H, Kumar S. Streptococcus intermedius Causing Necrotizing Pneumonia in an Immune Competent Female: A Case Report and Literature Review. Case Rep Pulmonol. 2016;2016:7452161. doi:10.1155/2016/7452161 REFERENCE #2: Huggins JT, Sahn SA. Causes and management of pleural fibrosis. Respirology. 2004 Nov;9(4):441-7. doi: 10.1111/j.1440-1843.2004.00630.x. PMID: 15612954 REFERENCE #3: Jackson A, Maybee J, Moran MK, Wolter-Warmerdam K, Hickey F. Clinical Characteristics of Dysphagia in Children with Down Syndrome. Dysphagia. 2016 Oct;31(5):663-71. doi: 10.1007/s00455-016-9725-7. Epub 2016 Jul 12. PMID: 27405422 DISCLOSURES: No relevant relationships by Timothy Barreiro, source=Web Response No relevant relationships by Rishanki Jha, source=Web Response No relevant relationships by Oyidiya Osoka, source=Web Response
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