Experience with Surgery for Thymoma
Associated with Pure Red Blood
Cell Aplasia*
Report
Shigefumi
Atsushi
and
of Three
Fujimura,
Yamauchi,
Tasuku
Nakada,
Cases
M.D.,
F.C.C.P.;
Takashi
Kondo,
M.D.;
Masashi
Hand..a,
M.D.;
M.D.,
M.D.;
F.C.C.P
We present
three cases ofthymoma
associated
with pure red
blood cell aplasia in which thymomectomy
and thymectomy
were performed.
Case 1, a patient
with pure red blood cell
aplasia
and hypogammaglobulmnemia,
was
treated
after
surgery
with immunosuppressive
agents.
She did not show
any remission
and died eight months
after the operation.
Case 2, a patient
with pure red blood cell aplasia
alone,
showed
transient
erythropoiesis
only in the early postoperative
period and died one year and seven months
after
the operation.
Patient
3 had pure red blood cell aplasia
T
hymoma
often
is associated
with
various
alone
before
surgery
and was treated
after surgery
with
prednisolone
and fluoxymesterone.
He showed good remission from the aplasia
after
these
freatments;
however,
myasthenia
gravis appeared
seven months
after the operation.
These
results
seem
to show that such combined
therapy
as applied
in case 3 may be effective
for some of the
patients
with pure red blood cell aplasia
and thymoma;
however,
the effects of thymomectomy
or thymectomy
(or
both) are still controversial
for the treatment
of pure
red
blood cell aplasia.
who
pure
autoim-
mune
diseases
during
its natural
history.’
Among
them,
myasthenia
gravis has been proven
to be caused
by circulating
antibody
to the acetylcholine
receptor,
and the
however,
protocol
for treatment
the causes
of other
the
Diseases
Supported
Manuscript
is being
established;
diseases
associated
with
Reprint
Department
of Surgery,
the
Research
Institute
CASE
CASE
astinum
x-ray
she
the
winter
of1974,
tive
cough,
and
1980,
admitted
1. Preoperative
roentgenograms
Case 1; center,
case 2; and
Left,
of patients
right,
case
with
thymomas
each
with
was readmitted
J une
Japan
FIGURE
aplasia.
woman
on a chest
hospitalized
17.
980,
treatment
for pure red
from a surgical
point
REPORTS
1
A 59-year-old
for Chest
and Cancer,
Tohoku
University,
Sendai,
Japan.
in part by Japan
Welfare
Ministry
Board
(58-32).
received
April 15, 1984; revision
accepted
January
requests:
Dt
Fujimura,
1-4-6-501
Katahira,
Sendai
surgery
for thymoma
associated
with
cell aplasia.
These
cases are presented
in order
to look for an effective
blood
cell aplasia
with thymoma
of view.
thymoma,
such
as pure
red blood
cell aplasia
and
hypogammaglobulinemia,
are not known
nor are their
effective
treatments.
Recently,
we had experience
with
three
patients
*From
underwent
red blood
to
a tumorous
film
at examination
had
time,
noticed
Tohoku
associated
shadow
frequently
she
tonsillitis
vertigo
received
University
with
In August
pneumonia
together
with
Medical
pure
red
right
medi-
in 1967.
and
In 1978,
1978,
Since
producshe
the
for two months.
where
was
patient
From
breathlessness;
Center,
blood
mass
fever
antibiotics.
days.
and
of the
ofa
developed
pneumoniafor40
with
she
had
she was
she
was
cell
3.
CHEST
Downloaded From: http://journal.publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21480/ on 06/15/2017
I 88 I 2 I AUGUST,
1985
221
Table
1-Results
ofPeripheral
ofPreoperative
Blood
and
Case
Data
Peripheral
1
Case
g/dl
Reticulocytes,
x 104/pJ
304
cells,
Erythroblasts,
percent
4.9
3.9
5.0
a,-globulin,
percent
12.2
9.9
12.0
3-globulin,
percent
7.9
7.6
8.2
-y-globulin,
percent
5.2
14.4
10.2
42.3
29.0
5,500
11,400
5.64
mg/dl
660
1, 140
830
mg/dl
60
103
159
40
27
183
47.9
40.5
11
percent
0
0
0
1gM,
0.6
Serum
percent
63.0
76.8
complement,
test,
Antinuclear
6.4
2.2
5.0
Myelocytes
22.0
18.6
13.8
ESR,
Metamyelocytes
11.0
20.6
16.0
C-reactive
Band
16.2
27.4
26.6
7.4
8.0
6.4
0.4
0.6
0.4
1.6
0.6
3.4
0.8
5.8
3.2
Promyelocytes
Segmented
percent
percent
percent
percent
percent
RBC,
Red
blood
diagnosed
as
cell
count;
having
and
for surgical
had
On
right
treatment
The
behind
red
patient
results
blood
cell
blood
cell
Since
February
in
the
peripheral
and
she
level,
composition
was
examinations
were
to pokeweed
protein
showed
The
measured
surface
with
cells
with
relatively
scant
(Fig
malignant
nature
After
surgery
died
ment
in the
eight
liver,
myocardium
section,
specimen
3).
This
The
there
slightly
on Feb
separated
predominantly
nuclei.
OKT-4,
percent
.
.
.
.
. .
43.2
OKT-8,
percent
.
.
.
. . .
38.2
.
.
.
.
12.8
protein;
spring
spindle-cell
thymoma
the
patient
was
months
and
spleen,
after
pancreas,
was
given
and
the
findings.
abundant
also
the
chest
cytes
to
was
operation
without
At autopsy
fat. There
and
thyroid
the
was
bone
diffuse
gland.
any
sud-
marrow
hemosiderosis
Brown
atrophy
3.8
by 7.6
with
soft
2
right
1). A blood
cell
10/p.l
no
and
revealed
plasma
rate.
chest
patient
x-ray
was
Medical
cell
Center
on
The
chest
mediastinal
count
component.
(control,
7.6
percent).
film
near
the
the
erythro-
(Table
and
1). On
a decrease
Other
examinations
Cutaneous
reactivity
immunoglobulin-positive
a
5, 1982.
x-ray
showed
reticulocytes
and
with
Jan
side
no erythroblasts
cell
film
transferred
aplasia
palpable.
the
in surface
tumor
was
by 4.2
cm.
portions
lymphocyte
thymomectomy
and
thymectomy
encapsulated.
The
specimen
well
On
cut
grossly
section,
to
cells
migration
(Fig
by
had
thin
fibrous
spindle-shaped
4). There
were
on Jan
measured
a parenchymal
it had
separated
epithelial
surface
septa
nuclei
(Fig
with
no malignant
2).
scant
patterns
in
specimens.
patient
received
showed
Finally,
no immunosuppressive
temporary
level
remission
was
the
patient
surgery.
up
a decrease
died
bone
to 13.2
in this
of cerebral
Autopsy
therapy
in
increased
however,
after
CASE
level
not
surgery
findings;
percent
the
at one
began
softening
was
after
marrow
to be
one
year
month
progres-
and
seven
performed.
3
right-sided
man
pleuritis
had
had
a history
been
ofpneumonia
in childhood.
noted
roentgenologically
loss ofappetite
and
Old
for
ten
years.
of
The
1983,
remarkable.
the
The
blood
was
on
underwent
The
A 58-year-old
had
and
percent
patient
1982.
surgery;
improve-
x
red
.
normal.
26,
these
spleen
marrow
cells
to 0.2
the
304
on
University
pure
(Fig
a decrease
thereafter.
patient
wall
nucleated
sive
agents
The
Tohoku
and
mass
bone
after
no
1981.
nor
number
contained
immunosuppressive
azathioprine.
liver
erythroblast
showed
in September
via
.
sedimentation
shadow
a tumorous
months
ACTH,
noted
erythrocytic
abnormal
ofthymoma
revealed
PPD
ESR,
An
department
Neither
in
and
of1981.
were
our
film
A 63-year-old
percent
total
The
were
1.0
85.0
by fibrous
Lymphocytes
88.0
47.3
.
but
a smooth
2,508
89.7
.
to
specimen
was
histologically.
hematologic
no erythroblasts,
encapsulated.
spindle-shaped
as prednisolone,
denly
well
On cut
1,722
9.5
lymphocyte
thymectomy
+
93.7
Histologically,
purified
dinitrochlorobenzene.
and
+
.
The
of other
to
+
.
in the
serum
blastogenesis
tests
70
.
levels
of
the
10
17
.
showed
marrow
results
skin
and
no
some
B-lympho-
2). The
inactive
portions
the
in
10
6,432
examination,
investigations
lymphocyte
(PPD)
soft
2). Histologically,
epithelial
(Table
was
were
was
bone
decrease
thymomectomy
mass
there
there
in the
are
without
immunoglobulins.
and
by 12.1 by 8.7 cm.
parenchymal
(Fig
(PWM)
tumorous
9.8
cells
as decreased
underwent
and
immunologic
decreased
on
patients
anemia
but
apparent
of tuberculin
patient
10, 1981.
also
reported
derivative
The
and
three
severe
blood,
as in serum
mitogen
in the
was
plasma
an
as well
and
mass
.
. .
percent
anterior
lymphocytosis,
and
nails
a tumorous
.
-
OKT-3,
anemia
1).
1, there
Biochemical
blood
-y-globulin
(Fig
peripheral
cells,
to disappear.
on the
revealed
examinations
relative
in nucleated
tended
sternum
1. In patient
with
a trichophytosis
film
I
-
10
(2#{176})
/p.l
OKIaI,
depart1978,
protein
-
20
B, percent
*TP
aplasia
(x )
antibodies
Count,
count.
to our
I
-
T, percent
0.4
was transferred
had
x-ray
of hematologic
in Table
decrease
patient
the
reticulocytes
the
white
direct/indirect
mm/hr
diagnosis
A chest
erythroblasts,
such
0
on Jan 19, 1981.
the
side
summarized
septa
0.2
CH5OU/ml
Lymphocytes
stomatitis.
examination,
cyte
2.0
pure
The
hepatosplenomegaly.
any
22.0
0.2
WBC,
thymoma,
hypogammaglobulinemia.
ment
16.0
0
29.0
perceni
cells,
Others,
CASE
mg/dl
Coombs’
67.8
1.80
IgA,
0
143
1.78
2.28
ratio
IgG,
6. 15
/p.l
Neutrophils,
the
22.6
X 10/p.l
0
Plasma
6.0
64.3
a1-globulin,
0
Lymphocytes,
6.1
64.0
3
0
percent
Monocytes,
6.2
69.5
Case
9.1
Myeloblasts,
Eosinophils,
2
0
Albumin-globulin
Megakaryocytes,
had
percent
Case
9.4
8,400
Nucleated
1
g/dl
Albumin,
292
marrow
Basophils,
Case
Data*
3
0
39.0
/i.i.l
of Preoperative
Examinations
6.8
percent
Platelets,
WBC,
in
Case
2
TP,
238
x104/i.l
Hemoglobin,
the
2-Results
Immunologic
Marrow
blood*
RBC,
Bone
Table
Examinations
Bone
patient
and
was
anemia
noted.
presented
with
associated
He
was
with
admitted
an abnormal
into
our
faintness
shadow
in August
on chest
department
on
x-ray
Sept
8,
1983.
man
developed
weakness
and
slight
breathlessness
Chest
roentgenographic
examination
222
Downloaded From: http://journal.publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21480/ on 06/15/2017
Surgery
revealed
a large
for Thymoma
tumorous
(Fujimura
at a!)
FIGURE
thelial
original
4. Thymoma
showing
densely
cells
with
scant
lymphocytes
magnification
x 100).
lymphocyte
level,
anti-T-cell
antibodies
The
patient
1983.
17,
to
category
8.4
had
the
by
was
had
no tumor
After
Gross
from
shadow
on
pleuritis
on theleft
anemia
without
erythroblasts
the
Biochemical
and
and
test
of
1 (top),
left
but
-y-globulins
Coombs’
features
case
side
2
behind
(middle)
the
(Fig
1). Investigations
any
reticulocytes
normal
findings
immunologic
each
was
and
external
case
kind
cut
and
anterior
chest
ofperipheral
(Table
examinations
FIGURE
3. Thymoma
showing
thelial
cells with lymphocytes
magnification
x 100).
There
was
wall,
blood
1).
There
ofgranulopoiesis
in bone
showed
ofimmunoglobulin
positive.
surface
of thymoma
case 3 (bottom).
(Table
a relative
with
were
levels
indirect
densely
packed
spindle-shaped
(case 1) (hematoxylin-eosin,
in the
the
operation.
period.
The
patient
no
breathlessness.
the
of
red
After
mechanism
B-
epioriginal
marrow
On
blood
cell
on the
12th
level
decreased
marrow
showed
erythropoiesis
was
in order
pure
red
the
204
X
the
we
and
Downloaded From: http://journal.publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21480/ on 06/15/2017
I 88
was
was
zero,
and
blood.
autoimmune
to stimulate
bone
therapy
with
started
FIGURE
5. Thymoma
showing
two separate
areas,
lymphocytes
and another
with
scant lymphocytes.
are spindle-shaped
(case 3) (hematoxylin-eosin,
tion
X 100).
but
peripheral
a presumed
aplasia
erythroblasts
of progressive
level
in the
days,
operation,
because
the
up to
five weeks
percent
reticulocyte
cell
going
postoperative
9.6
after
1O/p.l
in
were
to 24 percent
later,
simultaneously,
CHEST
an
epi-
there
improvement
19th
to suppress
blood
area
had
Many
levels
and
weeks
readmission
readmission,
in the
three
portion
5). Histologically,
rapid
five weeks
hospital
count
(Fig
Histo-
a dark
capsule.
reticulocyte
was discharged
2).
lymphocytes.
showed
this
Bone
to our
the
patient
78 percent
scant
be
surgical
measured
(Fig
light
to
its
specimen;
another
nuclei
into
peripheral
however,
readmitted
marrow.
2). The
and
at this
old
showed
normal
decrease
after
the
specimen
in the
on Nov
seemed
excised
and
invasion
but
encapsulated
areas
spindle-shaped
cell
thymectomy
thus,
well
with
by monoclonal
pericardium;
The
was
two dominant
findings;
respectively;
and
pattern
surgery
8 percent
and
of lymphocytes,
hematologic
2.
were
dominant
cells
and
capsule
lobe
epi-
range.
a thickened
thymoma.
cm
measured
normal
thymomectomy
upper
7.3
collections
thelial
FIGURE
removed
left
there
epithelial
within
had
invasive
11.9
logically,
subsets
the
tumor
was
by
lymphocyte
were
underwent
The
invasive
but
packed
spindle-shaped
(case
2) (hematoxylin-eosin,
one
with abundant
Epithelial
cells
original
magnifica-
I 2 I AUGUST,
1 985
223
moma,
temic
myasthenia
lupus
gravis,
erythroblastopenic
erythematosus
in one
anemia
patient.
Can
Med
and
Assoc
6 Hint
sys-
J
109:733-38
4 Cooper
A, Wells
a
JV. Pemphigus
in a patient
with
foliaceus,
serological
myasthenia
evidence
gravis,
ofSLE.
and
Aust
plasma
erythroblast
SB,
Kao
V. Studies
inhibitor
nuclei.
on red
to
heme
Proc
Nati
cell
aplasia:
1. demonstration
synthesis
Aced
Sci
and
USA
an
1967;
of
antibody
8
to
BH,
1967;
Manaligod
and
review
literature.
of the
Hayward
AR,
pression
by
The
syndrome
a review
pancytopenia
1982;
58:493-500
TI.
anemia:
Medicine
7 Rogers
J
NZ
1981; 11:277-80
5 Krantz
Robertson
reports.
thymoma
Med
E,
blastopenic
1973;
of thymoma
of 56
cases
JR.
Blazek
J
Am
Med
P, Webster
Paolucci
erythro-
three
case
46:225-64
WV.
Thymoma
hypogammaglobulinemia:
thymoma
and
including
1968;
ADB,
patient
associated
report
with
of a case
and
44:154-64
Kohier
lymphocytes.
P. Pre-B
Clin
Exp
cell
sup-
Immunol
48:437-42
Heart and Mind: Neurocardiology
Update
The University
ofKansas
Medical Centerwill
present
this one-day program
on September
7 at
the Hyatt Regency
Hotel,
Kansas City, Missouri.
For information,
contact
Ms. Jan Johnston,
Office ofContinuing
Education,
University
of Kansas Medical Center,
39th and Rainbow
Blvd,
Kansas
City, KS 66103 (913:5884480).
Pulmonary
The St. Paul-Ramsey
Paul, September
19-21.
Ramsey
Medical
Center,
Medical
For
Center,
and TB Update
will
information,
640 Jackson
present
contact:
Street,
this course
Continuing
St. Paul
55101
at the Radisson
Medical
Plaza Hotel,
Education,
St.
St.
Paul-
(612:221-3992).
CHEST/88/2/AUGUST,1985
Downloaded From: http://journal.publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21480/ on 06/15/2017
225
moma,
temic
myasthenia
lupus
gravis,
erythroblastopenic
erythematosus
in one
anemia
patient.
Can
Med
and
Assoc
6 Hint
sys-
J
109:733-38
4 Cooper
A, Wells
a
JV. Pemphigus
in a patient
with
foliaceus,
serological
myasthenia
evidence
gravis,
ofSLE.
and
Aust
plasma
erythroblast
SB,
Kao
V. Studies
inhibitor
nuclei.
on red
to
heme
Proc
Nati
cell
aplasia:
1. demonstration
synthesis
Aced
Sci
and
USA
an
1967;
of
antibody
8
to
BH,
1967;
Manaligod
and
review
literature.
of the
Hayward
AR,
pression
by
The
syndrome
a review
pancytopenia
1982;
58:493-500
TI.
anemia:
Medicine
7 Rogers
J
NZ
1981; 11:277-80
5 Krantz
Robertson
reports.
thymoma
Med
E,
blastopenic
1973;
of thymoma
of 56
cases
JR.
Blazek
J
Am
Med
P, Webster
Paolucci
erythro-
three
case
46:225-64
WV.
Thymoma
hypogammaglobulinemia:
thymoma
and
including
1968;
ADB,
patient
associated
report
with
of a case
and
44:154-64
Kohier
lymphocytes.
P. Pre-B
Clin
Exp
cell
sup-
Immunol
48:437-42
Heart and Mind: Neurocardiology
Update
The University
ofKansas
Medical Centerwill
present
this one-day program
on September
7 at
the Hyatt Regency
Hotel,
Kansas City, Missouri.
For information,
contact
Ms. Jan Johnston,
Office ofContinuing
Education,
University
of Kansas Medical Center,
39th and Rainbow
Blvd,
Kansas
City, KS 66103 (913:5884480).
Pulmonary
The St. Paul-Ramsey
Paul, September
19-21.
Ramsey
Medical
Center,
Medical
For
Center,
and TB Update
will
information,
640 Jackson
present
contact:
Street,
this course
Continuing
St. Paul
55101
at the Radisson
Medical
Plaza Hotel,
Education,
St.
St.
Paul-
(612:221-3992).
CHEST/88/2/AUGUST,1985
Downloaded From: http://journal.publications.chestnet.org/pdfaccess.ashx?url=/data/journals/chest/21480/ on 06/15/2017
225