Luo et al. Eur J Med Res (2021) 26:48 https://doi.org/10.1186/s40001-021-00519-5 European Journal of Medical Research CASE REPORT Open Access Pulmonary sequestration with Aspergillus infection presenting as massive hemoptysis and hemothorax with highly elevated carcinoembryonic antigen in pleural efusion that mimics advanced lung malignancy Wei Luo1, Tong‑chen Hu2, Lincheng Luo1 and Ya‑lun Li3* Abstract Background: Pulmonary sequestration (PS) associated with massive hemoptysis, hemothorax, and elevated tumor markers or even lung malignancy has been reported in several studies. These clinical features combined with lung lesions on chest imaging are sometimes hard to diferentiate from lung malignancies and often complicate the diag‑ nostic procedure. Case presentation: A 45‑year‑old man with PS presented with massive hemoptysis, hemothorax, and extremely elevated carcinoembryonic antigen (CEA) in pleural efusion was initially misdiagnosed with advanced lung carci‑ noma, but was ultimately diagnosed with PS with Aspergillus infection. Conclusions: PS is rarely concurrent with lung cancer; most of the time, it is misdiagnosed as a malignancy, espe‑ cially when presenting with a fungal infection, which could remarkably elevate CEA in pleural efusion. Keywords: Pulmonary sequestration, Aspergillus infection, Carcinoembryonic antigen Introduction imaging are hard to diferentiate from lung malignan- Pulmonary sequestration (PS) is a congenital pulmonary cies and often complicate the diagnostic procedure [5, malformation with an estimated incidence of 2.2–6.6%. 6]. Here, we describe a patient with massive hemoptysis, It can be defned as a part of the lung that has no nor- hemothorax, and remarkably elevated pleural fuid CEA mal communication with the bronchial tree and receives who was nearly diagnosed with primary lung malignancy blood supply from systemic arteries [1]. In recent dec- but was ultimately diagnosed with PS with Aspergil- ades, there have been several reports of PS associated lus infection. Written consent was obtained from our with massive hemoptysis, hemothorax, and elevated Institutional Review Board and the patient for this case tumor markers such as carcinoembryonic antigen (CEA) report. or even diagnosed with lung malignancy [2–4]. Tese clinical features combined with lung lesions on chest Case presentation A 45-year-old man presented with massive hemoptysis *Correspondence: [email protected] and breathlessness on exertion for 3 days with a history 3 Department of Respiratory and Critical Care Medicine, West China of 30 pack-years of smoking. He had moderate fever but Hospital of Sichuan University, Chengdu 610046, Sichuan, China no night sweats, anorexia, or weight loss. Te patient did Full list of author information is available at the end of the article © The Author(s) 2021. This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. 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Hemoptysis was pre-existing conditions that may cause these symptoms. stopped after bronchial arterial embolism (BAE) with Te patient had a respiratory rate of 28 breaths/min and coils and polyvinyl alcohol (Fig. 1b, c). Bronchoscopy oxygen saturation of 91% on ambient air. Chest physical and thoracoscopy were performed after BAE. Bronchos- examination revealed mild respiratory distress, obvious copy was unremarkable, and thoracoscopy showed some percussive dullness, and diminished breath sounds on lesions on the parietal pleura, bloody efusion and fbrous the left side of the chest. Other physical examination was adhesions (Fig. 1d). However, the histopathology of the unremarkable. He had moderate leukocytosis (16,500/ pleural lesion only implied chronic infammation with µL) and procalcitonin (11.3 mg/L), and serum CEA was some necrosis. CT-guided needle biopsy of the retro- normal (2.2 ng/mL). Other laboratory tests were nor- cardiac mass was performed twice (Fig. 1e). None of the mal. Enhanced chest computed tomography (CT) scans histopathological and cytological examinations suggested revealed a cystic mass that was mildly enhanced in the malignancy, but only elevated neutrophils and Aspergil- left lower lobe supplied by an anomalous artery arising lus hyphae emerged. Because there was no evidence of from the descending thoracic aorta with massive pleural malignancy, left lower lobectomy was performed, and efusion (Fig. 1a). Terefore, he was initially diagnosed the lesion was removed thoroughly (Fig. 1f). Postsurgi- with PS with infammatory pleural efusion. Neverthe- cal histopathology examination confrmed the diagno- less, tube thoracostomy revealed hemothorax, while the sis of intralobar pulmonary sequestrations (IPSs) and CEA in the pleural efusion reached 98 ng/ml, and cyto- the existence of septate hyphae as well as some fbrinoid logical analysis demonstrated that neutrophils were the necrosis (Fig. 1g, h). After the surgery, the CEA in the major type of leukocyte. Tese fndings complicated the pleural efusion and the temperature returned to normal. diagnosis, such that we could not rule out the possibil- Te postoperative recovery was unremarkable, and the ity of advanced stage lung cancer, so lobectomy was not patient was discharged 7 days later. One month later at Fig. 1 a Axial section of thoracic contrast‑enhanced CT showing a heterogeneous mass at the posterior basal segment of the left lower lobes with massive pleural efusion (black arrow). b Bronchial artery angiography showing an aberrant artery arising from the descending thoracic aorta (black arrow). c Aberrant artery embolized with coils and polyvinyl alcohol. d Thoracoscopy showing a lesion on the parietal pleura, bloody efusion, and fbrous adhesions (black arrow). e CT‑guided needle biopsy showing the needle in the mass. f Lobectomy showing the aberrant artery as well as the coils (black arrow and rectangle); the right upper corner shows an enlarged view of the artery and coil (green arrow). g Septate hyphae in the resected mass with a chronic infammatory reaction (black arrow and rectangle); the right upper corner shows an enlarged view of the septate hyphae (green arrow). H&E staining, original magnifcation 200. h Lobectomy showing fbrinoid necrosis with infammatory cell infltration. H&E × staining, original magnifcation 200 × Luo et al. Eur J Med Res (2021) 26:48 Page 3 of 4 outpatient follow-up, the patient showed good recovery, possibility. Tird, in PS patients with recurrent or fatal except for slight left chest pain. respiratory symptoms, surgery is the frst choice, even if malignancy cannot be ruled out. Discussion We describe a rare case of PS with Aspergillus infec- Conclusions tion that presented as massive hemoptysis, hemothorax, PS could be associated with elevated tumor markers, extremely elevated CEA in pleural efusion and initial especially lung Aspergillus infection, which could result radiological fndings mimicking advanced lung carci- in a remarkably increased CEA in either the pleural efu- noma, complicating the therapeutic strategy (not receiv- sion or serum, leading to a misdiagnosis of advanced lung ing surgery frst) and subjecting the patient to repeated malignancy. biopsies, unnecessary anxiety, and high medical costs. Tus far, only seven cases of lung cancer with PS have Acknowledgements been reported [7]. In contrast, up to 21% of PS cases have Not applicable. been misdiagnosed as lung cancer among 2625 cases over Authors’ contributions a 10-year period in China [8], and Matsuoka et al. [5] also WL, LL, and YL worked together for the treatment of this patient, including interventional surgery. TH performed the lobectomy. WL and YL conducted reported that PS tends to be misdiagnosed as lung can- the literature review and wrote and translated the manuscript. cer, especially with high levels of tumor markers. CEA is one of the most widely used tumor markers for Funding lung cancer and colorectal carcinoma, and can be pro- There is no funding support for this case. duced in the epithelium of the respiratory and digestive Availability of data and materials tracts. CEA participates in the innate immune defense All data and materials are available. system and has a role in cell adhesion [9]. An oncologic correlation worth mentioning is the relationship between Declarations the hemothorax and the remarkably elevated CEA in Ethics approval and consent to participate the pleural efusion in this case. Given the sensitivity Approval was obtained from our Institutional Review