Bilateral multiple pulmonary artery aneurysms associated with cavitary pulmonary tuberculosis: a case report

Background Pulmonary artery aneurysms constitute <1% of aneurysms occurring in the thoracic cavity. Congenital cardiac defects are responsible for the majority (>50%) of cases, however, pulmonary artery aneurysm is a rare sequelae of pulmonary tuberculosis reported in about 5% of patients with chronic cavitary tuberculosis on autopsy. The natural history of this potentially fatal condition remains poorly understood and guidelines for optimal management are controversial. Case presentation A 24-year-old man, a nursing student of African descent, was referred to us from an up-country regional hospital with a 4-week history of recurrent episodes of breathlessness, awareness of heartbeats and coughing blood 3 weeks after completing a 6-month course of anti-tuberculosis drugs. A physical examination revealed conjuctival and palmar pallor but there were no stigmata of connective tissue disorders, systemic vasculitides or congenital heart disease. An examination of the cardiovascular system revealed accentuated second heart sound (S2) with early diastolic (grade 1/6) and holosystolic (grade 2/6) murmurs at the pulmonic and tricuspid areas respectively. Blood tests showed iron deficiency anemia, prolonged bleeding time, and mild hyponatremia. A chest radiograph revealed bilateral ovoid-shaped perihilar opacities while a computed tomography scan showed bilateral multiple pulmonary artery pseudoaneurysms with surrounding hematoma together with adjacent cystic changes, consolidations, and tree-in-bud appearance. Our patient refused to undergo surgery and died of aneurismal rupture after 9 days of hospitalization. Conclusions The presence of intractable hemoptysis among patients with tuberculosis even after completion of anti-tuberculosis course should raise an index of suspicion for pulmonary artery aneurysm. Furthermore, despite of its rarity, early recognition and timely surgical intervention of pulmonary artery aneurysm is crucial to reducing morbidity and preventing the attributed mortality.


Case presentation
A 24-year-old man, a nursing student of African descent, was referred to us from an up-country regional hospital for further investigations and expert management. His past medical history was unremarkable and he denied any history of tobacco or intravenous drug use (IVDU), chest trauma, sexually transmitted infection (STI) or open tuberculosis (TB) contact. He was diagnosed with pulmonary TB based on constitutional symptoms and chest X-ray findings, and had completed a 6-month course of anti-TB medications (isoniazid, rifampicin, pyrazinamide, and ethambutol) 7 weeks prior his visit to our institution. He was somewhat symptom-free for about 3 weeks when he developed recurrent episodes of breathlessness, awareness of heartbeats and coughing blood, which had gradually worsened and persisted for about 4 weeks prior this index visit.
On examination, he was a sick-looking but oriented and well-kempt young man. He had a blood pressure of 92/57 mmHg, pulse rate of 121 beats/minute, respiratory rate of 19 breaths/minute and temperature of 36.7°C. His body mass index (BMI) was 21.2 kg/m 2 (weight 59 kg and height 1.67 m). A physical examination revealed conjuctival and palmar pallor but there were no stigmata of connective tissue disorders, systemic vasculitides or congenital heart disease. A respiratory system examination revealed bilateral symmetrical chest movements; however, dullness and reduced breath sounds were noted on the mammary and inframammary regions bilaterally on percussion and auscultation respectively. An examination of the cardiovascular system revealed accentuated second heart sound (S 2 ) with early diastolic (grade 1/6) and holosystolic (grade 2/6) murmurs at the pulmonic and tricuspid areas respectively.
Hematological and biochemical tests revealed iron deficiency anemia [hemoglobin (Hb) 8.18 g/dL, mean corpuscular volume (MCV) 61.8 fL, mean corpuscular hemoglobin (MCH) 19.2 pg/cell and red cell distribution width (RDW) 21.9%], prolonged bleeding time [prothrombin time (PT) 14.6 s and partial thromboplastin time (PTT) 32.7 s], and mild hyponatremia [sodium concentration (Na + ) 132 mmol/L]. Serological tests for human immunodeficiency virus (HIV), hepatitis B and C, and syphilis were all negative. Moreover, occult blood test was negative and an abdominal ultrasonography scan was unremarkable. A sputum culture revealed Pseudomonas aeruginosa 3++ sensitive to ciprofloxacin and gentamicin. A chest X-ray showed bilateral, ovoidshaped perihilar opacities extending to lower lung zones with loss of silhouette sign on the left cardiac border (Fig. 1). An electrocardiogram (ECG) displayed a sinus tachycardia while echocardiography (ECHO) was evident for right ventricular hypertrophy, mild tricuspid regurgitation with estimated right ventricular systolic pressure (RVSP) of 29 mmHg (that is, mild pulmonary hypertension) and multiple focal dilatations along the right and left pulmonary arteries. Systolic functions were, however, preserved (ejection fraction 53%). A GeneXpert test was positive. A computed tomography (CT) scan of his chest showed bilateral multiple pulmonary artery pseudoaneurysms with surrounding hematoma together with adjacent cystic changes, consolidations and tree-in-bud appearance (Figs. 2, 3, 4, and 5). The aneurysms on the right pulmonary artery measured 3.81×2.55 cm and 6.96×5.34 cm whereas those on the left measured 2.61×2.03 cm, 6.85×4.45 cm and 7.05×4.03 cm respectively. His main pulmonary artery (2.55 cm), ascending (2.28 cm), arch (1.89 cm), and descending aorta (2.00 cm) had normal caliber and dimensions. We entertained a diagnosis of bilateral multiple pulmonary pseudoaneurysms associated with cavitary pulmonary tuberculosis and admitted him to the general medical ward. Intravenous ciprofloxacin 500 mg 12 hourly, bisoprolol 5 mg once daily, tadalafil (40 mg) and ferrous sulfate (270 Fig. 1 Chest X-ray (posteroanterior view) displaying bilateral, ovoidshaped perihilar opacities extending to lower lung zones with loss of silhouette sign on the left cardiac border mg) plus folic acid (300 mcg) were initiated. Moreover, our patient was counseled regarding surgery and prognosis of his condition; however, he refused to undergo any surgical procedure. After 9 days of hospitalization, our patient died of aneurismal rupture. Due to religious beliefs, relatives of the deceased refused an autopsy and one was not done.
Hemoptysis in a tuberculosis (TB) setting is relatively common, usually self-limiting or controlled by anti-TB drugs. Nevertheless, presence of a massive or several episodes of minor hemoptysis in TB is life-threatening, likely originating from the arterial system, and require early and aggressive intervention [39,40]. Rasmussen's aneurysm is a rare sequalae of pulmonary TB resulting from gradual weakening of the pulmonary artery wall from the adjacent tubercular cavity leading to thinning and pseudoaneurysm formation [36][37][38][39]. It has been reported in approximately 5% of autopsy series involving patients with chronic cavitary tuberculosis [39][40][41][42][43][44]. Despite absence of clear guidelines for treatment of PAA, several  surgical techniques including aneurysmorrhaphy, Dacron graft replacement, pulmonary allograft replacement, and replacement with combination of Dacron prosthesis and stentless bioprosthesis are advocated [9,19,21,28,45,46]. Furthermore, patients presenting with massive hemoptysis are usually treated with a bronchial artery embolization procedure [47]. However, such an embolization procedure technique requires special care especially in pseudoaneurysms as they are easily prone to rupture with resultant fatal bleeding [39,43,47].
Our patient refused to undergo any surgical procedure and consequently died of aneurismal rupture on the ninth day of hospitalization. Arguably, even if he had opted for surgery, the presence of multiple bilateral large pseudoaneurysms would have potentiated a high risk of fatal bleeding, among other complications, using any surgical technique. Nevertheless, watchful waiting, as in this case, portends a very poor prognosis. To the best of our knowledge, this was the first ever case of TBassociated PAA to be diagnosed and documented in Tanzania. We hope that upon its publication this case will sensitize practitioners to consider and assess for PAA whenever faced by hemoptysis not responding to otherwise routine therapy.

Conclusions
In conclusion, presence of intractable hemoptysis among TB patients even after completion of an anti-TB course should raise an index of suspicion for PAA. Furthermore, despite its rarity, early recognition and timely surgical intervention is crucial in reducing morbidity and preventing the attributed mortality. Ethics approval and consent to participate Ethical clearance was granted by the Directorate of Research of the Jakaya Kikwete Cardiac Institute Consent for publication Written informed consent was obtained from the patient's next-of-kin for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Competing interests
The authors declare that they have no competing interests.

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