(Chest. 2000;118:269-270.)
© 2000
American College of Chest Physicians
Unilateral Diaphragmatic Paralysis Following Bronchial Artery Embolization for Hemoptysis*
Sally A. Chapman, MD;
Mark D. Holmes, MD and
D. James Taylor, MD
*
From the Royal Adelaide Hospital Chest Clinic, Adelaide, Australia.
Correspondence to: Sally A. Chapman, MD, Royal Adelaide Hospital Chest Clinic, 275 North Terrace, Adelaide 5000, Australia; e-mail: schapman{at}mail.rah.sa.gov.au
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Abstract
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Bronchial artery embolization is an effective treatment
for patients with hemoptysis. Serious complications are rare, but may
occur if the arterial supply to other structures is compromised. We
present a case of unilateral diaphragmatic paralysis following
bronchial artery embolization in a patient with cystic fibrosis. We
believe that the diaphragmatic paralysis was due to the inadvertent
obstruction of the left pericardiacophrenic artery during the
embolization procedure, with compromise of the phrenic nerve blood
supply. This resulted in a significant loss of lung function in our
patient, who did not recover despite the subsequent return of
diaphragmatic function.
Key Words: bronchial arteries • cystic fibrosis • diaphragm • embolization • hemoptysis • phrenic nerve • therapeutic
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Introduction
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Bronchial
artery embolization is a well-accepted and effective form of treatment
for massive and recurrent hemoptysis. Overall, the success rate for
control of active hemoptysis is 90%; however, recurrence of hemoptysis
is seen in up to 27% of cases.1
The most commonly
reported complication is that of chest pain, which occurs in as many as
90% of cases. Other reported complications are rare and are related to
the inadvertent compromise of the arterial supply to other organs. Such
complications include spinal cord infarction, esophagobronchial
fistula, and cerebrovascular accident. 1
We present a case
of hemoptysis treated with bronchial artery embolization, which was
complicated by the development of unilateral diaphragmatic paralysis
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Case Report
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A 29-year-old woman with cystic fibrosis and severe cystic
bronchiectasis presented with recurrent hemoptysis. There was a 2-year
history of minor hemoptysis < 50 mL in association with exacerbations
of her bronchiectasis, and there was an episode of recurrent hemoptysis
of between 50 and 200 mL for 4 days that had resolved with rest and IV
antibiotics.
She presented with further hemoptysis, having hemorrhages of between
200 and 300 mL on 3 consecutive days, and underwent bronchial
angiography and embolization. Angiography of the right lung showed that
the upper two thirds was supplied by a large, tortuous bronchial artery
with numerous abnormal branching vessels. This bronchial artery was
embolized with polyvinyl alcohol particles and two 5-mm steel coils,
with complete occlusion of its lumen and cessation of distal flow.
Angiography of the left lung showed that the upper left lung was
supplied by a tortuous bronchial artery that was being fed from both
the aorta and internal mammary artery. The feeding vessels were both
partially occluded by the placement of polyvinyl alcohol particles in
both vessels and by a single 5-mm steel coil in the aortic origin. Flow
was reduced, but abnormal blood vessels remained. Further embolization
was deferred due to the long duration of the procedure. After the
procedure, she developed right-sided pleuritic chest pain that lasted
for 4 days.
Recurrent hemoptysis of between 50 and 100 mL developed 2 months later
and failed to settle with conservative therapy. Further bronchial
artery angiography and embolization were arranged. During this
procedure, the left bronchial artery arising from the internal mammary
artery was embolized using numerous polyvinyl alcohol particles.
Complete occlusion of the abnormal vessels was achieved, and completion
aortic images showed only very small residual vessels supplying the
lungs. She was discharged from hospital 2 days postprocedure and was
apparently well at that time.
On review 2 weeks later, she described increased dyspnea since the
bronchial artery embolization. Lung function testing showed a marked
decline in FEV1, from 1.17 to 0.67 L, and also in
FVC, from 1.75 to 1.05 L. Her chest radiograph showed a raised left
hemidiaphragm. A fluoroscopic study showed paradoxical movement of the
left hemidiaphragm, suggesting paralysis. Phrenic nerve studies
confirmed a conduction block of the left phrenic nerve consistent with
infarction. Review of the angiography images showed that the left
pericardiacophrenic artery had been occluded at the embolization
procedure (Fig 1 , 2)
.
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Figure 1.. Angiography of the left internal mammary artery
before embolization, showing the abnormal left bronchial artery (small
arrow) and the left pericardiacophrenic artery descending along the
left heart border (large arrow). The distal internal mammary artery is
poorly visualized due to diversion of blood flow to the abnormal
bronchial artery.
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Nine months later, she noted improved exercise tolerance. An
improvement in her lung function was seen, with a rise in
FEV1 to 0.87 L and in FVC to 1.36 L. A chest
radiograph showed normal diaphragmatic position, and fluoroscopy showed
normal diaphragmatic movements.
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Discussion
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Recent studies have shown that major hemoptysis in people with
cystic fibrosis can be safely and effectively treated with bronchial
artery embolization, with immediate control of hemoptysis in 84 to
100% of those treated.2
3
4
Apart from chest pain, fever,
and transient dysphagia, no significant complications have been
reported in these studies.
Unilateral diaphragmatic paralysis is often asymptomatic, but up to
24% of people with this condition experience persistent shortness of
breath,5
particularly if they have underlying lung disease
or obesity. Orthopnea is frequently reported, and oxygen desaturation
may be seen on exercise or when asleep. Our patient experienced
significant dyspnea, particularly on exertion, but had no orthopnea. No
oxygen desaturation was detected on overnight oximetry.
Unilateral diaphragmatic paralysis is usually due to phrenic nerve
dysfunction, which is often idiopathic in origin. In cases in which a
cause is identified, the most common etiologies are malignant
infiltration due to bronchogenic carcinoma, postsurgical complications
of neck or thoracic surgery, trauma, infection, and neurological
disease.5
Following coronary bypass surgery, between 8%
and 17% of patients are estimated to have some degree of unilateral
diaphragmatic paralysis, with higher incidences seen in patients
receiving internal mammary artery grafts.6
The phrenic nerve is accompanied by the pericardiacophrenic artery in
the thorax. This is one of the first branches of the internal mammary
artery. Studies in animals have confirmed that the phrenic nerve blood
supply is largely derived from this artery, with perfusion of the left
phrenic nerve reduced by 71% following left internal mammary artery
dissection with ligation of this branch.6
We think that the left phrenic nerve infarction and consequent left
diaphragmatic paralysis observed in our patient were secondary to the
inadvertent obstruction of the left pericardiacophrenic artery, during
the embolization of the left bronchial artery feeder from the left
mammary artery. This had serious consequences in our patient, with
significant loss of lung function, which failed to return to baseline
despite subsequent diaphragmatic recovery. To our knowledge, this
complication of bronchial artery embolization has not been described
previously. We believe that awareness of this complication and
identification of the pericardiacophrenic artery before the
embolization of abnormal bronchial arteries arising from the internal
mammary artery should enable this complication to be avoided.
Received for publication September 7, 1999.
Accepted for publication December 30, 1999.
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References
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Cohen, AM, Doershuk, CF, Stern, RC (1990) Bronchial artery embolization to control hemoptysis in cystic fibrosis. Radiology 175,401-405[Abstract/Free Full Text]
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Sweezey, NB, Fellows, KE (1990) Bronchial artery embolization for severe hemoptysis in cystic fibrosis. Chest 97,1322-1326[Abstract/Free Full Text]
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Cipolli, M, Perini, S, Valletta, EA, et al (1995) Bronchial artery embolization in the management of hemoptysis in cystic fibrosis. Pediatr Pulmonol 19,344-347[ISI][Medline]
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Piehler, JM, Pairolero, PC, Gracey, DR, et al (1982) Unexplained diaphragmatic paralysis: a harbinger of malignant disease? J Thorac Cardiovasc Surg 84,861-864[Abstract]
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O’Brien, JW, Johnson, SH, VanSteyn, SJ, et al (1991) Effects of internal mammary artery dissection on phrenic nerve perfusion and function. Ann Thorac Surg 52,182-188[Abstract]
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Abstract
Introduction
