Pulmonary Arteriovenous Fistula Showinga Fall in Shunt Fraction

rhythm was restored after five days of therapy. Propranolol
therapy
also has been used successftilly to treat SVT in a fetus with
Wolff-Parkinson-White syndrome by Teuscher et al.9 Hypo
glycemia and bradycardia have been observed by Habib and
transplacental drug transfer and impaired neonatal drug disposi
McCarthy'6
j@ four neonates
with good Apgar
scores
whose
during
pregnancy,
labor and delivery:
tion. J Pediatr 1977; 91:812
18 Rubin PC. Beta blockers in pregnancy.
305:1323
evidence
for
N EngI J Med 1981;
mothers had received propranolol during pregnancy. Cottrill
et al'7 have also reported marked bradycardia and hypo
gIycemia in a neonate whose mother had received 160 mg of
propranolol daily. These authors have considered propran
olol therapy during pregnancy as a risk factor for the neonate.
However, Rubin,'8 on the basis ofcurrent available informa
tion on @3-blockers
in pregnancy, has cast doubts on the
adverse effects ofpropranolol on the fetus.
In conclusion, this case confirms the usefulness of ultra
sound examination in the diagnosis offetal cardiac failure and
the effectiveness and safety oftransplacental digoxin therapy
forSVT
-
1 Kleinman CS, Donnerstein RL, DeVore GR, Jaffee CC, Lynch
DC, Berkowitz RL, et al. Fetal echocardiography for evaluation
ofin utero congestive heart failure. N EngI J Med 1982; 306:568
2 Newburger JW, Keane JR Intrauterine supraventricular
tachy
cardia. J Pediatr 1979; 95:780
3 Klein AM, Holzman IR, Austin EM. Fetal tachycardia prior to
the development of hydrops—attempted pharmacologic car
dioversion:
case report.
Am J Obstet
Hajime Maeda, M.D.; Yasumasa Monden,
M.D.;
Kazuya Nakahara, M.D.; Shinichiro Miyoshi, M.D.;
and Yasunaru Kawashima, M.D. , FC.C.P
A 23-year-oldmanwith pulmonaryarteriovenousfistulasof
the right middle lobe is described. During the incremental
exercise test, the shunt fraction dropped from 19 percent to
12 percent as the cardiac output increased. We discuss the
REFERENCES
4 Kerenyi TD, Gleicher
Pulmonary Arteriovenous Fistula
Showinga Fallin Shunt Fraction
DuringExercise*
Gynecol
1979; 134:347
N, Meller J, Brown E, Stienfeld
L,
mechanism ofthis fall in shunt fraction in this patient during
exercise.
E
xertional dyspnea is the most common symptom in
patients with a pulmonary arteriovenous (AV) fistula,
and the arterial oxygen tension has been reported to de
crease during exercise in about half the patients.'2 The
patient described herein showed a fall in shunt fraction
during the incremental
exercise test. His arterial
oxygen
Chitkara U, et al. Transplacentalcardioversionof intrauterine
tension first increased and then decreased. The purpose of
supraventricular tachycardia with digitalis. Lancet 1980;2:393-5
this report is to describe the behavior of the fistulas and the
other pulmonary capillaries of this patient in response to
increased cardiac output durng exercise.
5 Lingman
C, Ohrlander
S, Ohlin P Intrauterine
digoxin treat
ment offetal paroxysmal tachycardia. BrJ Obstet Gynaecol 1980;
87:340
6 Harrigan JT, Kangos JJ, Sikka A, Spisso KR, Natarajan N,
CASE REPORT
Rosenfeld D, et al Successful treatment offetal congestive heart
failure secondaryto tachycardia.N Engl J Med 1981;304:1527
7 Wiggins W, Clewell W, Bowes W, Johnson M, Appariti K, Wolfe
RR. Successful diagnosis and therapy ofarrhythmias,
congestive
heart failure in the fetus with digoxin (Abstract). Pediatr Cardiol
1982; 2:175
8 WolfF, BreukerKH, SchlenskerKA, BolteA. Prenataldiagnosis
and therapy offetal heart rate anomalies; with a contribution on
the placental transfer ofverapamil. J Perinat Med 1980; 8:203
9 Teuscher A, Bossi E, Imhof P. Erb E, Stocker FP, Weber JW,
et al Effect of propranolol on fetal tachycardia in diabetic
pregnancy. Am J Cardiol 1978; 42:304
10 Rudolph AM, Heymann MA. Fetal and neonatal circulation and
respiration. Ann Rev Physiol 1974. 36:187
11 Hilrich
LM, Evrard
JR. Supraventricular
tachycardia
in the
newbornwithonsetin utero. AmJ Obstet Gynecol1955;70:1139
12 Nadas AS, Daeschner CW, Roth A, Blumenthal SL. Paroxysmal
tachycardia in infants and children. Pediatrics 1952; 9:167
13 Berman W, Ravenscroft PJ, Sheiner LB. Heymann MA, Melmon
KL, Rudolph AM. Differential effects of digoxin at comparable
concentrations in tissues offetal and adult sheep. Circ Res 1977.
41:635
14 Chan V, Tse TF, Wong V. Transfer ofdigoxin across the placenta
and into breast milk. Br J Obstet Gynaecol 1978; 85:6059
15 Rogers MC, Willerson JT, Goldblatt A, Smith TW Serum
digoxinconcentrationsin the humanfetus,neonateand infant.N
Engl J Med 1971; 287: 1010
16 Habib A, McCarthy JS. Effects on the neonate of propranolol
administered
during pregnancy.
J Pediatr 1977; 91:808
17 Cottrill CM, McAllister RG, Gettes L, Noonan JA. Propranolol
A 23-year-old
man was referred
to our hospital for evaluation
of
occasional mild dyspnea while sleeping. He had never felt dyspnea
on exertion. There was no history ofpulmonary disease or congenital
malformations
smoking.
in the patient's
family, and he had no history
of
The patient was a well-developed young man with normal vital
signs and no cyanosis or digital clubbing.
There were no hemangio
mas or telangiectasias. The heart sound was normal and auscultation
of the lungs revealed no rales or bruits.
The hemoglobin level was 17.9. The ECG findings were normal.
The chest x-ray film showed two rounded densities in the right lung.
Pulmonary
angiographic
studies (Fig 1) demonstrated
two pulmo
nary AVfistulas originating from the vessels ofthe right middle lobe.
The pulmonary function tests were normal.
He underwent the incremental exercise test3on a cycle ergometer
(Mijnhardt
Medical Instrument,
Model FEM5)one
week before and
four months after surgery. Minute ventilation (yE), oxygen con
sumption
(Vo@), and carbon
dioxide
production
(Vco@) were
mea
sured by an on-line microcomputer combined with a hot-wire
respiratory
flow meter,4 a zirconia solid electrolyte
oxygen analyzer,'
and an infrared carbon dioxide analyzer (Minato Medical Science,
System RM-200). The radial artery was cannulated,
and a Swan
Ganz catheter was inserted for arterial and mixed venous blood
sampling. The cardiac output and right-to-left shunt fraction were
calculated according to the following formulas:
*From the First Department of Surgery, Osaka University Medical
School, Fukushima-ku,
Osaka, Japan.
Reprint requests: Dr Maeda, First Department of Surgery, Osaka
University Medical School, Fukushima-ku, Osaka, Japan
CHEST I 85 I 4 I APRIL, 1984
Downloaded From: http://publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21406/ on 06/15/2017
575
16,-
14
12
10
.—.• Preop.C
0.
h--i
Preop.Shunt
0—C
Posto@, C 0.
E5••-tt@6
Posto@,Shunt
4
U
2
---@-
0
-A
-
I
I
Supine
I
Sitting
1
2OWatts
50 Watts
75W.tts
100Watts
Body Position and Work Rate
FIGURE
2.
Changes
in cardiac
output
(CO)
and
shunt
relating
to
body position and work rate.
DISCussIoN
The clinical triad of cyanosis, exertional dyspnea, and
digital clubbing is a frequent finding in patients with a
pulmonary AV fistula.6 The severity ofthe clinical symptoms,
however, depends on the degree of shunt—lO to 56 percent
ofthese patients have been reported to have no complaints. 7.8
Some
papers―29 have described
the influence
of exercise
on patients with a pulmonary AV fistula, but no case has
reported a fall in shunt fraction during exercise. Slutter
Eringa et al' performed ergometric studies on 20 patients
with a pulmonary
FIGuRE
1.
originating
@
from the vessels ofthe
showingtwoarteriovenous
fistulas
was 6. 1 L/min,
and the shunt fraction
was 19 percent.
and reported
fistula associated with chronic obstructive
(COPD)
and ischemic
heart disease.
right middle lobe.
cardiac output
‘¿@°2
(Ca02 —¿
CVO@)
shunt fraction
(Cc'O2—CaO@) I (Cc'O2—C@'O@)
where Ca02 = arterial oxygen content; C@tO2
mixed venous
oxygen content; and Cc'02 = end-capillary oxygen content. The
end-capillary oxygen tension was regarded to be identical with the
alveolar oxygen tension. All studies were performed with the patient
breathing room air. The results of the tests are shown in Figures 2
and 3. In the preoperative evaluation, the intracardiac and pulmo
nary arterial pressures were normal. At rest in the sitting position,
the arterial oxygen tension (PaO@.)was 73.1 mm Hg, the cardiac
output
@
Pulmonaryangiogram
AV fistula,
that eight of them
showed a decrease in arterial oxygen saturation (Sa02) of
more than 5 percent and/or a decrease in Pa02 of more than
10 mm Hg. Harrow et a12reported a case of pulmonary AV
pulmonary
disease
In that case, the
exercise induced even greater hypoxemia and shunting (49 to
43 mm Hg and 37 to 39 percent, respectively). Bye et al9
described
a 7-year-old
boy and the results
of exercise
tests
before and after surgery, but they did not calculate the shunt
25@
:s Preop.
20
o—o
Postop.
During
the incremental
exercise test in the sitting position, the shunt
remained almost constant as the cardiac output increased, so the
shunt fraction dropped inversely to 12 percent. The Pa02 increased
to 79.9 mm Hg at 20 W and then decreased to 70.7 mm Hg at 50 W.
The test stopped at this workload because the patient complained of
skipped heart beat and precordial discomfort.
On Jan 24, 1983, he underwent
right middle lobectomy. His
postoperative course was uneventful.
Four months after surgery, he was admitted for reevalutation.
Pulmonary angiography
demonstrated
no residual fistulas. He
underwent the exercise test again in the same manner. At rest, the
Pa02 was 102.4 mm Hg, the cardiac output was 4.3 IJmin, and the
shunt fraction was 2.8 percent. During exercise test, the shunt
fraction
did not exceed 4 percent.
The preoperative
and
postoperative values of VE, Vo2 and Vco2 were almost the same.
15
‘¿@ 10
(It
Supine
S.tt@ng
2OWatts
Body Position
FIGURE
3.
Changes
in shunt
S0Watts
75Watts
100 Watts
and Work Rate
fraction
relating
to body
position
and
work rate.
576
Downloaded From: http://publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21406/ on 06/15/2017
PulmonaryArteriovenousFistula(Maedaet a!)
fraction.
In our patient,
the shunt
fraction
as the cardiac output increased.
dropped
during
exercise
Studies ofpulmonary
func
tion tests and cardiac catheterization
of this patient
were
normal. The theoretic
mechanism
ofthis fall ofshunt fraction
is as follows.
In the normal
person,
pulmonary
vascular
resistance
is lowered in response to increased
cardiac output
by the mechanism
of recruitment
and distention
of the
@
pulmonary
vascular
exercise,
We considered
resistance,
except
but the resistance
that the increased
blood flow circulated
normal
capillaries.
pulmonary
that pulmonary
for the fistulas, dropped
during
ofthe fistulas did not change, so
only through
This would
explain
the
why the
results differed from those of the case of Harrow et al2, who
had a larger fistula and combined with COPD.
The PaO2increased at 20 W but decreased at 50 W despite
the reduction in shunt fraction. At that time, the alveolar
oxygen
tension
decreased
from
110.6 mm
Hg to 102.6 mm
Chronic Mountain Sickness at an
Elevationof 2,000 Meters*
Christian Gronbeck ill, Maj, MC, USA, FCC.?
A resident living at Lake Tahoe, Calif, at an elevation of
2,000 meters, had fatigue, edema, and erythrocythemia.
Hematocrit was 63 percent, and arterial blood gas values
revealed hypoxemia and respiratory acidosis. Results of
pulmonary function tests, sleep study, and thyroid function
all were normal. Erythrocytosis, cor pulmonale, and respi
ratory
acidosis
resolved
after the patient
moved
to sea level.
This patient suffered from chronic mountain sickness. Her
symptoms resolved with relief of hypoxia.
C
hronic mountain sickness (Monge's disease) is commonly
described in Leadville, Col, and Cero de Pasco, Peru. 1.2
Hg, and the mixed venous oxygen tension decreased from
Lake Tahoe, Calif, with an altitude ofonly 2,000 m, has not
40.0 mm Hg to 31.0 mm Hg. We consider
previously
that the decrease
of
the Pa02 at higher work load resulted from these two facts.
We cannot definitely explain the reason why the patient
complained ofmsld dyspnea while asleep. The shunt fraction
been
considered
may have increased
in the left-side-up
recumbent
position,
but we regret not having measured
it at such a position.
CASE
The results in our patient show the value of the study of
shunt
fraction
and cardiopulmonary
cise to clarify the mechanism
patient
with a pulmonary
dynamics
of adaptation
during
exer
to exercise in a
AV fistula.
REFERENCES
1 Slutter-Eringa
H, One NGM, Slutter HJ. Pulmonary
teriovenous fistula. Am Rev Respir Dis 1969; 100:177-88
ar
in health
ofhuman
adaptation
REPORT
A 67-year-old woman was admitted to Letterman Army Medical
Center for evaluation offatigue and erythrocytosis. The patient was a
nonsmoker residing at Lake Tahoe, Calif. at an elevation of2,000 m
(6,500 ft). She had chronic medical problems of obesity, hyperten
sion, and adult onset diabetes mellitus for which she took hvdro
chlorothiazide and insulin.
Six months prior to admission, she was seen elsewhere for gradual
onset offatigue, dyspnea on exertion, and a weight gain oflS.9 kg (35
Ib). Two months later, she noted pedal edema for which she was
treated with triamterene
(Dyrenium) with modest improvement.
2 Harrow EM, Beach PM, Wise JR. Lynch C, Graham WGB,
Wright C. Pulmonary arteriovenous fistula. Chest 1978; 73:92-94
3 Wasserman K, Whipp BJ. Exercise physiology
disease. Am Rev Respir Dis 1975; 112:219-49
an outpost
to high altitude. I report a case ofchronic mountain sickness
in a 67-year-old woman living at Lake Tahoe. Her hypercap
nia and erythrocytosis resolved when she moved to sea level.
and
One week prior to admission to Letterman, her blood pressure was
160/95 mm Hg, and her hematocrit reading was 63 percent. The
chest was clear, and a cardiovascular examination result was normal.
She had pitting edema to both knees. She was given furoseinide
(Lasix), 80 mg/day for four days, and referred to this hospital.
On admission to Letterman Army Medical Center, a physical
S. A bidirectional
principle. J Appl
examination revealed no change except resolution of the pedal
edema. Chest x-ray films showed the heart size at the upper limit of
normal. Complete blood cell count showed a hemoglobin of 19.1
g/dl, hematocrit of6O.2 percent, and normal indices, platelets. and
5 Elliott SE, Seggen FJ, Osborn JJ. A modified oxygen gauge for
the rapid measurement ofPo, in respiratory gases. J Appl Physiol
1966;21:1672-4
leukocytes. Arterial blood gases showed a pH of7.44, Pco2 of49 mm
Hg, and PO@of6O mm Hg on room air. The serum sodium level was
4 Yoshiya I, Nakajima T, Nagai I, Jitsukawa
respiratory
flowmeter using the hot-wire
Physiol 1975; 38:360-5
6 Moyer JH, Grantz C, Brest AN. Pulmonary
fistulas. Am J Med 1962; 32:417-35
arteriovenous
142 mEq/L; potassium, 5.4 mEq/L; chloride, 100 mEq/L; and CO2.
32 mEq/L. Serum creatinine and BUN levels were normal, and
blood glucose level was 304 mg/dl. Thyroid function tests were
normal, and thyroid-stimulating
hormone was normal at 4 lU/mi.
The ECG was normal.
7 Sloan RD, Cooley RN. Congenital pulmonary arteriovenous
aneurysm.
AJR 1953; 70:183-210
8 Dines I)E, Arms BA, Bernatz PE, Comes MR. Pulmonary
arteriovenous
fistulas. Mayo Clin Proc 1974; 49:460-5
9 Bye MR. Cern)' FJ, Gingell RL. Increased exercise capability
after repair of a pulmonary arteriovenous
fistula. Chest 1982;
82:373-5
10 GrazierJB, HughesJMB,
MaloneyJE, WestJB. Measurements
ofcapillary dimensions and blood volume in rapidly frozen lungs.
J Appl Physiol1969;26:65-76
Sleep study results were normal with no sleep apnea. Pulmonary
function tests showed poor effort on spirometric study. The FE\T,
was 1.45 L (80 percent), FVC was 1.52 L(59 percent), and exhalation
time was less than two seconds. Total lung capacity by the neon
dilution single-breath method was 3.23 L (76 percent), and Dsb was
169 percent. Voluntary hyperventilation
decreased the Pco2 from
45.8 to 30.5 mm Hg. Echocardiography
showed the right ventricle
size to be at the upper limit ofnormal, and a multigated uptake scan
8From Pulmonary Medicine Service, Department
of Medicine,
Letterman Army Medical Center, Presidio of San Francisco, CA.
The opinions or assertions contained herein are the private views of
the authors and are not to be construed as official or reflecting the
views ofthe Department ofthe Army or the Department of I)efense.
CHEST I 85 I 4 I APRIL, 1984
Downloaded From: http://publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21406/ on 06/15/2017
577