Repeat Episodes of Massive Hemoptysis Due to an Anomalous Origin of the Right Bronchial Artery in a Patient with a History of Coccidioidomycosis

Blerina Asllanaj, MD

Elizabeth Benge MD

Yi McWhworter DO

Sapna Bhatia MD

Department of Internal Medicine

HCA Healthcare

Mountain View Hospital

Las Vegas, NV, USA

Abstract

Anomalous bronchial arteries originate outside the space bound by the T5 and T6 vertebrae at the major bronchi. Here, we highlight a case of a 37-year-old man with a past medical history of coccidioidomycosis and who presented with massive hemoptysis. A bronchial angiogram showed the patient had a right bronchial artery originating anomalously from the left subclavian artery. The patient ultimately underwent a bronchial artery embolization, after which he achieved symptomatic remission.

Introduction

Hemoptysis from primary coccioidomycosis is unusual and should prompt a search for other causes (1). These could include bronchitis, malignancy, or rarely, a fungus ball. Anomalous bronchial arteries have origins outside the space bound by the T5 and T6 vertebrae at the level of the major bronchi (2). Bronchial artery embolization is the standard treatment for patients with ruptured anomalous bronchial arteries and resultant hemoptysis (3). Here, we present a unique case of a 37-year-old male with a past medical history of coccidioidomycosis and previous episodes of massive hemoptysis who was found to have an anomalous right bronchial artery originating in his left subclavian artery. Symptomatic remission was achieved with bronchial artery embolization. To our knowledge, this is the only reported case of a patient with a history coccidioidomycosis and a ruptured anomalous right bronchial artery that was successfully treated with bronchial artery embolization.

Case Presentation

Our patient is a 37-year-old man with a past medical history significant for coccidioidomycosis (resolved nine years prior) and previous episodes of massive hemoptysis who presented to our emergency room with multiple episodes of hemoptysis over the course of one day. On admission, he reported a five-pack year smoking history. He denied hematemesis, dyspnea, and angina, a history venous thromboembolism and alcohol and recreational drug use.

In the emergency department, the patient was afebrile, his blood pressure was 177/119 mmHg, heart rate was 96 beats/min, respiratory rate was 16 breaths/minute, and his oxygen saturation was 95% on room air. The patient’s physical exam revealed diffuse rales throughout the right lung and decreased breath sounds in the right lower lobe. The remainder of the patient’s physical exam was negative for acute abnormalities.

His lab values on admission were significant only for an elevated D-dimer at 1.28 mcg/mL; his hemoglobin was 14.2 gm/dL and his INR was 0.93 sec/mL. His chest radiograph showed ill-defined patchy parenchymal densities over the bilateral lower lobes (Figure 1).

Figure 1. Chest x-ray reveals ill-defined patchy parenchymal densities over the lower lobes suggest evolving multifocal pneumonia or atypical viral pneumonia.

He experienced a witnessed episode of hemoptysis, expectorating 300 cc’s of blood, prompting an emergent bronchoscopy. During the bronchoscopy, bloody secretions were noted to in his right lower lobe. A five centimeter dark red gelatinous material was removed and sent for pathology studies alongside bronchoalveolar lavage washings. Two mL’s of 2% epinephrine were administered, after which no active oozing was noted. The patient was then intubated for airway protection and admitted to the intensive care unit.   

A repeat chest radiograph revealed opacification throughout the right lung with evidence of volume loss (Figure 2).

Figure 2. Chest x-ray showing interval development of opacification throughout the right lung with evidence of volume loss including rightward mediastinal shift. The left lung is clear.

The patient was empirically treated for atypical pneumonia with azithromycin, ceftriaxone, dexamethasone, and albuterol breathing treatments. A computed tomography angiogram (CTA) of the chest with contrast showed multifocal flocculent and nodular infiltrate posterolateral aspect right lower lobe as well as mild mucous plugging and bronchial edema. Bronchial angiography confirmed the branching of the right bronchial artery from the left subclavian artery (Figure 3) and evidence of shunting to the right lower lobe (Figure 4).

Figure 3. Bronchial angiography prior to embolization- right bronchial artery directly arising from the left subclavian artery and is unusually large in caliber.

Figure 4. Bronchial angiography confirms opacification of the right lower lobe.

After the aberrant artery was confirmed on bronchial angiogram, the patient underwent a right bronchial artery embolization. He was subsequently extubated. Pathology and bronchoalveolar lavage studies revealed blood; the patient’s infectious and autoimmune work-up were entirely negative. He was discharged home with self-care. To date, the patient has only experienced one episode of hemoptysis status-post embolization.

Discussion

Differential diagnoses for massive hemoptysis include pulmonary infections, such as coccidioidomycosis, invasive aspergillosis and Mycobacterium tuberculosis, and cardiovascular causes, including anomalous origin of bronchial arteries. A thorough diagnostic evaluation is needed to identify the causative underlying pathology, site of bleeding, and vascular anatomy, so that the appropriate treatment can be initiated (3).

Common origins of the bronchial arteries include the inferior aortic arch, distal descending thoracic aorta, subclavian artery, brachiocephalic trunk, thyrocervical trunk and coronary artery (5). A bronchial angiogram was pivotal in the evaluation of the anatomy of the bronchial arteries in our patient’s case, as it allowed for the optimal artery embolization due to the identification of an anomalous artery early in his treatment course.

The bronchial arteries can become dilated and tortuous due to chronic inflammatory diseases such as bronchiectasis, coccidioidomycosis and tuberculosis, and are prone to vascular remodeling; rendering them fragile (6). The new collateral vessels have thin walls, making them prone to rupture and bleeding. In our patient’s case, chronic inflammation related to his prior coccidioidomycosis infection contributed to the remodeling of his anomalous right bronchial artery, rendering it prone to rupture and therefore the likely culprit of his massive hemoptysis.

Conclusion

Overall, this case emphasizes the importance of recognizing the fragility of anomalous bronchial arteries. A history of previous episodes of hemoptysis can alert clinicians to the possibility of a congenital abnormality exacerbated by subsequent infection.   

References

1. Galgiani JN, Ampel NM, Blair JE, et al. 2016 Infectious Diseases Society of America (IDSA) Clinical Practice Guideline for the Treatment of Coccidioidomycosis. Clin Infect Dis. 2016 Sep 15;63(6):e112-46. [CrossRef] [PubMed]
2. Battal, B., Saglam M, Ors F et al. Aberrant right bronchial artery originating from right coronary artery–MDCT angiography findings. Br J Radiol. 2010;83(989): e101–e104. [CrossRef] [PubMed]
3. Keller FS, Rosch J, Loflin TG, Nath PH, McElvein RB. Nonbronchial systemic collateral arteries: significance in percutaneous embolotherapy for hemoptysis. Radiology. 1987 Sep;164(3):687-92. [CrossRef] [PubMed]
4. Ittrich H, Bockhorn M, Klose H, Simon M. The Diagnosis and Treatment of Hemoptysis. Dtsch Arztebl Int. 2017 Jun 5;114(21):371-381. [CrossRef] [PubMed]
5. Hartmann IJ, Remy-Jardin M, Menchini L, Teisseire A, Khalil C, Remy J. Ectopic origin of bronchial arteries: assessment with multidetector helical CT angiography. Eur Radiol. 2007 Aug;17(8):1943-53. [CrossRef] [PubMed]
6. Kathuria H, Hollingsworth HM, Vilvendhan R, Reardon C. Management of life-threatening hemoptysis. J Intensive Care. 2020 Apr 5;8:23. [CrossRef] [PubMed]

Cite as: Asllanaj B, Benge E, McWhworter Y, Bhatia S. Repeat Episodes of Massive Hemoptysis Due to an Anomalous Origin of the Right Bronchial Artery in a Patient with a History of Coccidioidomycosis. Southwest J Pulm Crit Care. 2021;23(3):89-92. doi: https://doi.org/10.13175/swjpcc037-21 PDF 

February 2017 Pulmonary Case of the Month

Abdalla Fadda, MD

Phoenix VA and Banner University Medical Center Phoenix

Phoenix, AZ USA

History of Present Illness

A 45-year-old man presented with weight loss, copious amounts of light green sputum, low grade fever and chest discomfort on the right. He had moved to Arizona 8 months ago. Two months later he developed hemoptysis and had increased cough with copious phlegm. He denied any fever, chills, malaise or fatigue.

Past Medical History, Social History and Family History

He has a history of tuberculosis in 2010 treated with 4 drug therapy for a year. The tuberculosis was not drug resistant. He had been treated with a 6-month course of voriconazole about 2 years ago.

Physical Examination

He was afebrile and his vital signs were unremarkable. He had decreased breath sounds in his right lower chest.

Laboratory

His CBC, electrolytes and urinalysis were unremarkable.

Chest Radiography

His admission chest x-ray is shown in Figure 1.

Figure 1. Admission PA of chest.

In regards to the chest x-ray which of the following are true? (Click on the correct answer to proceed to the second of six pages)

1. There are cavities in the right lung
2. There is a large right pleural effusion
3. There is volume loss in the right lung
4. 1 and 3
5. All of the above

Cite as: Fadda A. February 2017 pulmonary case of the month. Southwest J Pulm Crit Care. 2017;14(2):45-53. doi: https://doi.org/10.13175/swjpcc005-17 PDF

January 2017 Pulmonary Case of the Month

Jamie Bering, MD

Lewis J. Wesselius, MD

Department of Pulmonary Medicine

Mayo Clinic Arizona

Scottsdale, AZ

History of Present Illness

The patient is a 53-year-old woman transferred for acute respiratory failure and hemoptysis. She has a prior history of antiphospholipid syndrome and recurrent diffuse alveolar hemorrhage (DAH). She was admitted to another hospital about 2 weeks prior to transfer with hypoxic respiratory failure which ultimately required intubation. Bronchoscopy revealed a bloody aspirate raising concerns for recurrent DAH. She was started on high-dose solumedrol and extubated after 4 days. One week later, her respiratory status decompensated and her chest x-ray showed worsening diffuse bilateral opacities concerning for recurrent DAH. She was transferred to the Mayo Clinic Arizona for further evaluation. Upon arrival, she required 50% FiO2 by face mask to maintain adequate oxygenation and was started on broad-spectrum antibiotics. Her corticosteroids were tapered to 20 mg prednisone daily.

Past Medical History, Social History and Family History

She has a history of a mitral valve replacement with a St. Jude’s mechanical mitral valve and was on chronic anticoagulation with warfarin. In addition, there was a history of moderate aortic stenosis with moderate aortic insufficiency.

She had a history of diffuse alveolar hemorrhage, antiphospholipid antibody syndrome and possible systemic lupus erythematosus.

Medications

• Dapsone 100mg daily
• Ethacrynic acid 75mg daily
• Gabapentin 900mg QHS
• Lisinopril 20mg daily
• Meropenem 1g Q8 hrs
• Metoprolol 50 mg BID
• Prednisone 20mg daily
• Simvastatin 40mg QHS
• Vancomycin 1.5g Q12 hrs
• Warfarin 4mg T,F; 3mg SMWRSa

Physical Examination

• Vitals: T 36.3^◦ C; HR 79 beats/min; BP 100/63 mm Hg; RR 26 breaths/min; SpO2 99% face mask
• Gen: no acute distress
• HEENT: hematoma on chin
• Lungs: clear to auscultation and percussion
• Cardiac: Mechanical valve click

Laboratory

• CBC: WBC 15,900 cells per microliter (mcL); Hemoglobin 9.1 g/dL; hematocrit 29%; platelet count 156,000 cells per microliter.
• Electrolytes: within normal limits.
• BUN and creatinine: within normal limits.
• Blood sugar: 220 mg/dL.

Radiography

Her initial chest x-ray is shown in Figure 1.

Figure 1. Initial chest radiograph.

Which of the following best describes the chest x-ray? (Click on the correct answer to proceed to the second of four pages)

1. Diffuse lung consolidation
2. Previous median sternotomy
3. Previous mitral valve replacement
4. 1 and 3
5. All of the above

Cite as: Bering J, Wesselius LJ. January 2017 pulmonary case of the month. Southwest J Pulm Crit Care. 2016;14(1):1-5. doi: https://doi.org/10.13175/swjpcc146-16 PDF

June 2016 Pulmonary Case of the Month

Katie Murphy, MB BCh BAO¹

Henry D. Tazelaar, MD²

Laszlo T. Vaszar, MD³

¹Departments of Internal Medicine, ²Laboratory Medicine and Pathology and ³Pulmonary Medicine

Mayo Clinic Arizona

Scottsdale, AZ USA

Pulmonary Case of the Month CME Information

Members of the Arizona, New Mexico, Colorado and California Thoracic Societies and the Mayo Clinic are able to receive 0.25 AMA PRA Category 1 Credits™ for each case they complete. Completion of an evaluation form is required to receive credit and a link is provided on the last panel of the activity. 

0.25 AMA PRA Category 1 Credit(s)™

Estimated time to complete this activity: 0.25 hours

Lead Author(s): Katie Murphy, MB.  All Faculty, CME Planning Committee Members, and the CME Office Reviewers have disclosed that they do not have any relevant financial relationships with commercial interests that would constitute a conflict of interest concerning this CME activity.

Learning Objectives:
As a result of this activity I will be better able to:

1. Correctly interpret and identify clinical practices supported by the highest quality available evidence.
2. Will be better able to establsh the optimal evaluation leading to a correct diagnosis for patients with pulmonary, critical care and sleep disorders.
3. Will improve the translation of the most current clinical information into the delivery of high quality care for patients.
4. Will integrate new treatment options in discussing available treatment alternatives for patients with pulmonary, critical care and sleep related disorders.

Learning Format: Case-based, interactive online course, including mandatory assessment questions (number of questions varies by case). Please also read the Technical Requirements.

CME Sponsor: University of Arizona College of Medicine at Banner University Medical Center Tucson

Current Approval Period: January 1, 2015-December 31, 2016

Financial Support Received: None

History of Present Illness

A 77-year-old gentleman presented with 6 weeks of:

• Sinus congestion
• Bloody nasal discharge
• Cough with maroon sputum
• Dyspnea
• Hearing loss
• Painful peripheral neuropathy
• Left median neuropathy and left foot drop
• Fevers

Past Medical History, Social History and Family History

• No significant past medical history
• Retired
• Does not smoke
• Family history is noncontributory

Physical Examination

• Temperature of 37.8º C
• Bloody nasal discharge
• Lungs clear to auscultation and percussion
• Heart with a regular rhythm without murmur
• Neurologic findings consistent with his complaints

Laboratory Evaluation

• Elevated white blood cell count with a left shift
• Na+ 130 mEq/L
• 10-20 RBCs on urinalysis

Radiographic Evaluation

Initial chest x-day is shown in Figure 1.

Figure 1. Initial PA radiograph of chest.

Which of the following is (are) the next appropriate steps in the evaluation? (Click on the correct answer to proceed to the second of five panels)

1. Transthoracic echocardiogram
2. Treat with macrolide antibiotics for outpatient pneumonia
3. Thoracic CT scan
4. 1 and 3
5. All of the above

Cite as: Murphy K, Tazelaar HD, Vaszar LT. June 2016 pulmonary case of the month. Soutwest J Pulm Crit Care. 2016 Jun;12(6):205-11. doi: http://dx.doi.org/10.13175/swjpcc041-16 PDF

Traumatic Hemoptysis Complicating Pulmonary Amyloidosis

Erwan Oehler, MD¹

Charlotte Courtois, MD² 

Florent Valour, MD¹

¹Department of Internal Medicine

²Department of Pulmonary Medicine

French Polynesia Hospital Center

98716 Pirae, Tahiti

French Polynesia

Case Presentation

We present a 74-year-old man admitted to hospital for a fall occurring at home. His past medical history included histologically-proven pulmonary amyloidosis followed for fifteen years (Figure 1A), without involvement of other organs.

Figure 1A. Frontal chest radiography shows bilateral confluent, somewhat nodular and dense-appearing opacities with a background of faint linear and reticular opacities.

At admission, he complained of left chest pain related to a rib fracture (Figure 1B, arrow).

Figure 1B. Detail radiograph of the left upper thorax shows a fracture (arrow) of a posterolateral rib, superimposed on the background of dense-appearing linear and nodular parenchymal disease.

The next day, he presented with moderate hemoptysis, prompting performance of thoracic CT (Figure 1C and D) which showed a cavity filled with material of soft tissue attenuation.

Figure 1C and D. Axial thoracic CT displayed in soft tissue windows shows extensive bilateral nodular hyperattenuating tissue consistent with alveolar septal / diffuse pulmonary parenchymal amyloidosis. A cystic lesion with internal, dependent soft tissue attenuation (arrow, D) is present, consistent with a hematoma.

This soft tissue-filled cavity was located at the same level as the rib fracture, surrounded by calcified tissue, and presumably reflected a pulmonary parenchymal hematoma resulting from traumatically induced laceration of the inelastic calcified lung tissue.

Discussion

Pulmonary amyloidosis is a rare disease resulting from the extracellular deposition of insoluble fibrillar proteins aggregating in a β–pleated sheet configuration (1). Amyloidosis is classified according to the chemistry of the amyloid protein as AA secondary amyloidosis (SAA protein) -often related to chronic inflammatory disease- AL amyloidosis (monoclonal immunoglobulin light chains of the lambda or kappa type)-secondary to B lymphoproliferative disorders-and hereditary or familial amyloidosis (transthyretin and gelsolin). Dialysis-associated amyloidosis (βR₂R microglobulinemia) and “senile” amyloidosis SAA (wild-type transthyretin) are also recognized. Pulmonary amyloidosis may occur in three forms: tracheobronchial, nodular parenchymal and alveolar septal / diffuse parenchymal patterns (2). The two first forms (which include primitive pulmonary amyloidosis) are often remain localized to the respiratory system, whereas the alveolar septal / diffuse parenchymal form of amyloidosis, whose prognosis is more severe, often presents in a systemically. Parenchymal amyloid nodules grow slowly and generally remain asymptomatic but patients may also present with dyspnea, cough, hemoptysis or recurrent pneumonia (3).

References

1. Chu H, Zhao L, Zhang Z, Gui T, Yi X, Sun X. Clinical characteristics of amyloidosis with isolated respiratory system involvement: A review of 13 cases. Ann Thorac Med. 2012 (4):243-9. [CrossRef] [Pubmed]
2. Gilmore JD, Hawkins PN. Amyloidosis and the respiratory tract. Thorax. 1999;54:444-51. [CrossRef] [PubMed]
3. Vieira IG, Marchiori E, Zanetti G, Cabral RF, Takayassu TC, Spilberg G, Batista RR. Pulmonary amyloidosis with calcified nodules and masses - a six-year computed tomography follow-up: a case report. Cases J. 2009;2:6540. [CrossRef] [PubMed]

Cite as: Oehler E, Courtois C, Valour F. Traumatic hemoptysis complicating pulmonary amyloidosis. Southwest J Pulm Crit Care. 2015;11(4):173-5. doi: http://dx.doi.org/10.13175/swjpcc133-15 PDF

October 2012 Pulmonary Case of the Month: Hemoptysis from an Uncommon Cause

Lewis J. Wesselius, MD

Department of Pulmonary Medicine

Mayo Clinic Arizona

Scottsdale, AZ

History of Present Illness

A 39 year old woman is seen with a history of cough intermittently productive of small amounts of blood or blood-tinged sputum for 4 months. She reports no other respiratory symptoms and has otherwise felt well.

PMH, FH and SH

There was no significant PMH and no prior history of lung disease. Her father has a history of Parkinson’s disease and osteosarcoma. She is a nonsmoker, does not drink alcohol, and has never abused drugs. She has 2 children and is engaged to be remarried.

Physical Examination

Her physical examination is normal.

Chest X-ray

Her chest x-ray is below (Figure 1).

Figure 1. Panel A: Frontal chest radiography. Panel B: Lateral chest radiography.

Laboratory Evaluation

Hemoglobin was 13.2 g/dL and WBC was 8400 cells/μL with a normal differential. Urinanalysis was unremarkable.

Which of the following statements regarding hemoptysis is or are true?

1. A normal chest x-ray makes a benign cause of the hemoptysis more likely
2. Most patients with lung cancer are asymptomatic
3. Hemoptysis in children is usually associated with an infection or a foreign body
4. 1 + 3
5. All of the above

Reference as: Wesselius LJ. October 2012 pulmonary case of the month: hempotypsis from an uncommon cause. Southwest J Pulm Crit Care 2012;5:169-75.  PDF