Pulmonary Paragonimiasis Mimicking Tuberculosis in a Middle-Aged Burmese Female.

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22 Φεβ 2014 (πριν από 3 χρόνια και 3 μήνες)

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Pulmonary Paragonimiasis Mimicking Tuberculosis in a Middle
-
Aged Burmese Female.


Amjad Ali,
MD¹ , Allen T. Griffin, MD,¹ Hanan Farghaly, MD,² and Forest W. Arnold, DO, Msc¹


1
Division
of Infectious Diseases, University of
Louisville
.

2
Department
of Pathology, University of Louisville
, Louisville
, KY





Paragonimiasis

is

a

food
-
borne

parasitic

infection

resulting

from

ingestion

of

undercooked

crustaceans
.

Whi l e

uncommon

in

the

United

States,

Paragonimus

species

are

endemic

in

North

America,

South

America,

Asia,

and

Africa
.


Pulmonary

symptoms

are

most

common,

but

cerebral,

dermatologic,

and

abdominal

variations

are

recognized
.

Di a g n o s i s

is

supported

by

positive

serology

and

a

typical

exposure

history
.

P a t h o l o g i c

c o n f i r m a t i o n

by

visualization

of

eggs,

however,

is

preferred
.

In

the

subsequent

case

presentation,

we

elucidate

an

instance

of

pulmonary

paragonimiasis

mimicking

tuberculosis

in

a

middle
-
aged

Burmese

female



A

51
-
year
-
old

Burmese

female

was

admitted

to

the

hospital

for

cough

and

hemoptysis
.

S h e

r e l a y e d

a

h i s t o r y

of

hyperlipidemia

and

mild

chronic
-
obstructive

pulmonary

disease

(COPD),

but

was

otherwise

healthy
.

Upon

her

current

hospital

presentation,

she

described

having

a

non
-
productive

cough

intermittently

for

two

years,

which

had

been

attributed

to

COPD
.



However,

during

the

past

month,

the

cough

had

become

more

forceful

with

production

of

sputum

and

blood
.

S h e

c o u l d

not

recall

specific

circumstances

that

exacerbated

the

cough

and

noted

no

post
-
tussive

emesis
.

Associated

signs

and

symptoms

of

the

current

illness

included

night

sweats,

shortness

of

breath

on

exertion,

malaise,

periodic

fevers

to

101
°
F,

and

weight

loss

of

10

pounds

in

the

previous

month
.


On

one

occasion

since

becoming

ill,

the

patient

had

been

diagnosed

with

bronchitis

by

her

primary

care

physician

and

treated

with

doxycycline,

but

her

symptoms

did

not

abate
.

On

the

day

of

hospital

admission,

she

was

brought

to

the

emergency

room

after

an

episode

of

syncope

associated

with

coughing
.


The

patient

was

being

treated

with

low
-
dose

inhaled

corticosteroids

for

COPD

associated

with

smoking

and

baclofen

for

intermittent

muscle

spasms
.

She

reported

no

medication

allergies
.

Past

surgeries

included

a

tubal

ligation

13

years

earlier
.

She

was

a

30

pack

year

smoker,

but

consumed

no

alcohol

or

illicit

drugs
.


She

resided

in

the

city

of

Louisville,

Kentucky,

with

her

two

teen
-
age

children

as

well

as

three

other

adults

from

Burma
.

The

pati ent

worked

in

a

laundromat

and

immigrated

from

Burma

three

years

prior
;

however,

she

would

occasionally

return

to

her

home

country

to

visit

family

where

she

would

consume

uncooked,

freshwater

crustaceans,

including

crayfish

and

crab
.

Al t hough

she

was

not

certain,

she

could

not

recall

direct

exposure

to

individuals

with

tuberculosis

and

had

never

been

incarcerated
.

On

physical

examination,

the

patient

was

alert,

but

appeared

uncomfortable
.

H e r

t e m p e r a t u r e

w a s

98
.
0
°

F,

pulse

61

beats

per

minute,

blood

pressure

117
/
72

mm

of

Hg,

and

respiratory

rate

16

breaths

per

minute
.

Oxygen

saturation

was

98

%

on

ambient

air
.


The

oral

mucosa

was

dry,

but

revealed

no

lesions

or

thrush
.

There

was

no

cervical,

supraclavicular,

axillary,

or

inguinal

lymphadenopathy
.

The

cardi ac

exam

was

without

murmurs
.

Diffuse

wheezing

was

detected

on

the

pulmonary

exam

with

focal

rales

in

the

right

lower

lung

zones

noted
.



The

remainder

of

the

physical

exam

was

unremarkable
.

Laboratory

findings,

including

a

complete

blood

count

and

complete

metabolic

panel,

were

unrevealing,

with

the

exception

of

a

slight

eosinophilia

of

9

%

and

an

erythrocyte

sedimentation

rate

of

66

mm/hour

(Table

1
)
.




The

patient

had

a

negative

enzyme
-
linked

immunoabsorbent

assay

(ELISA)

for

human

immunodeficiency

virus

and

a

negative

QuantiFERON
®

TB

Gold

(
Cellestis
,

Ltd
.
,

Carnegie,

Australia)
.

Chest

r oent genogr am

r eveal ed

a

r i ght

l ower

l obe

i nf i l t r at e

t hat

cor r esponded

to

an

ovoid

area

of

mass
-
like

consolidation

on

computed

tomography

(CT)

in

the

right

lower

lobe

measuring

5
.
3

cm

x

3
.
2

cm

in

greatest

transverse

diameter

and

approximately

5
.
8

cm

craniocaudal

with

suspicion

of

early

cavitation

(Fig
.

1
)
.

M e d i a s t i n a l

and

hilar

lymphadenopathy

were

noted

as

was

a

partially

calcified

linear

lesion

emanating

from

the

diaphragm

(Fig
.

2
)
.

The

patient

was

suspected

to

have

a

subacute

community
-
acquired

pneumonia

(CAP)
.

She

was

felt

unlikely

to

be

infected

with

organisms

causing

acute

CAP
;

therefore,

antibiotics

were

not

initiated
.

The

predominant

differential

diagnostic

considerations

were

tuberculosis,

non
-
tuberculous

mycobacteria,

Nocardia

or

Actinomyces

infection,

or

less

likely

an

endemic

fungal

infection

contracted

in

the

United

States

or

Burma
.

S p u t u m

w a s

i n d u c e d

to

ascertain

the

etiology

of

her

illness
.

St a i n s

f o r

a c i d

f a s t

b a c i l l i

a n d

f u n g i

we r e

n e g a t i v e,

as

were

cultures

for

these

organisms
.

Standard

bacterial

cultures

yielded

normal

flora
.

A

CT
-
guided

lung

biopsy

was

pursued

for

definitive

diagnosis,

and

the

biopsied

tissue

was

submitted

for

microscopic

evaluation

as

well

as

cultures
.

P a t h o l o g i c

e x a m i n a t i o n

of

the

biopsied

tissue

revealed

scattered

eggs

embedded

in

the

lung

parenchyma,

producing

an

eosinophil
-
rich,

dense

inflammatory

reaction

with

nodular

granulomatous

lesions
.

The

visualized

eggs

[
90
-
100

µm

x

50
-
60

µm

(length

x

width)]

were

ovoid

and

contained

exudate

and

debris

(Fig
.

3
A)
.

A

Paragonimus

parasitic

infection

was

suspected

morphologically

and

was

further

supported

using

a

polarized

lens
.

The

visualized

eggs

were

birefringent

under

polarized

light,

a

distinctive

characteristic

of

Paragonimus

species

(Fig
.

3
B)
.

Polymerase

chain

reaction

testing

for

unequivocal

species

identification

was

not

undertaken,

and

details

of

ova

morphology

were

partially

distorted

on

processing
.

However,

based

on

the

size

of

the

ova

and

suspected

region

of

acquisition,

Paragonimus

westermani

was

thought

to

be

the

etiology

in

this

instance
.

Speci al

st ai ns

wer e

per f or med

on

the

lung

biopsy

for

acid

fast

bacilli

and

fungi
;

these

stains,

as

well

as

cultures

performed

for

these

organisms,

were

all

negative
.

As

the

diagnosis

was

secured

pathologically,

serology

for

Paragonimus

was

not

procured
.

Given

the

diagnosis

of

paragonimiasis,

the

patient

was

treated

with

praziquantel

at

25

mg/kg

three

times

per

day

for

two

days

and

discharged

from

the

hospital
.

She

was

followed

in

clinic

within

one

month,

and

she

reported

a

demonstrable

improvement

in

symptoms

In

summary
:



our

patient

presented

with

progressive

cough

and

hemoptysis

and

was

discovered

to

have

a

cavitary

infiltrate

on

chest

CT

with

a

worm

migration

track
.



Pathologic

examination

of

lung

tissue

revealed

ova

diagnostic

of

pulmonary

paragonimiasis

likely

contracted

from

eating

undercooked,

freshwater

crustaceans

in

the

patient’s

native

country

of

Burma
.



Similar

to

many

cases

of

paragonimiasis,

the

current

case

closely

resembled

pulmonary

tuberculosis

and

emphasizes

the

need

for

a

careful

review

of

dietary

habits

and

meticulous

pathologic

examination

of

tissue

to

ascertain

the

correct

diagnosis
.

1.
Procop

G. North American paragonimiasis (caused by
Paragonimus
kellicotti
) in the context of global Paragonimiasis.
Clin

Microbiol

Rev.
2009;22:415
-
446.

2.
Hu W. Studies on the life cycle of Paragonimus
heterotremus
.

Zhongguo

Ji

Sheng Chong
Xue

Yu
Ji

Sheng Chong Bing
Za

Zhi
. 1998;16:347
-
352.

3.
Diaz J. Boil before eating: paragonimiasis after eating raw crayfish in the
Mississippi River
Basin.
J La State Med Soc
. 2011;163:261
-
266.



.




Figure 3:
Paragonimus

egg embedded within a granulomatous
inflammation, 40X (Fig. 3A). The polarized lens highlights the
Paragonimus

egg’s
refractile

wall, 40 X (Fig. 3B). Eggs are 95 µm x
55 µm (
length x width).

BACKGROUND

CASE REPORT

CASE REPORT (Cont’d)

DISCUSSIONS (Cont’d
)

DISCUSSIONS

REFERENCES

CASE REPORT (Cont’d)

The

Paragonimus

genus

is

of

the

Trematode

class

within

the

phylum

Platyhelminthes

and,

thus,

is

closely

related

to

the

schistosomes
.

While

the

Paragonimus

genus

may

contain

nearly

40

distinct

species,

only

a

few

are

routinely

found

in

man,

the

most

common

of

which

worldwide

is

Paragonimus

westermani

(P
.

westermani
)
.

R e g i o n a l

d i f f e r e n c e s

e x i s t

w i t h

r e s p e c t

to

the

species

routinely

encountered,

with

the

most

frequently

diagnosed

in

indigenous

cases

in

the

United

States

being

Paragonimus

kellicotti

(P
.

kellicotti
)
.
1




Paragonimus

species

are

found

in

Asia,

Africa,

and

North

and

South

America
.
1

Th e

p r o p e n s i t y

to

acquire

the

disease

is

less

a

function

of

endemicity

than

regional

dietary

customs
.

E g g s

r e l e a s e d

f r o m

m a m m a l i a n

h o s t s

t y p i c a l l y

h a t c h

and

mature

into

miracidia

that

are

ingested

by

mollusks

such

as

snails
.

Wi t h i n

t h e

s n a i l,

t h e

mi r a c i d i a

e v e n t u a l l y

ma t u r e

i n t o

c e r c a r i a e
.

S u b s e q u e n t l y,

w h e n

t h e

s n a i l

is

consumed

by

a

crustacean,

cercariae

are

released

and

encyst

in

the

crustacean

as

metacercariae
.

Upon

consumption

of

the

crustacean

by

a

mammal,

metacercariae

excyst
,

penetrate

the

duodenum

of

the

mammalian

host,

travel

through

the

peritoneal

cavity,

and

cross

the

diaphragm

to

encyst

in

the

lungs

where

they

mature

into

adults
.
2

Thus,

consumpti on

of

uncooked

crustaceans,

crayfish
3

or

freshwater

crabs,
4

a

more

common

practice

in

Asia

and

Africa,

results

in

paragonimiasis
.



The

signs

and

symptoms

of

paragonimiasis

are

usually

pulmonary,

as

metacercariae

typically

encyst

and

mature

into

adults

in

the

lungs
.

The

t ypi cal

present at i on

i nvol ves

cough

and

hemoptysis

with

the

variable

presence

of

fever

and

other

systemic

complaints
;
5

however,

if

infection

is

limited

to

the

pleura,

hemoptysis

and

cough

can

be

absent,

and

pleuritic

chest

pain

may

dominate

the

clinical

picture
.
6

El evat ed

l evel s

of

blood

eosinophils

and

immunoglobulin

E,

though

common,

are

not

invariably

present
.
1


While

pleural

effusions,
6

pneumothoraces,
7

and

chylothoraces
8

have

all

been

noted,

the

most

common

radiologic

features

are

cystic

pulmonary

lesions,

nodules,

or

cavitary

lung

lesions
.
8
,
9

Li near

opaci t i es

represent at i ve

of

trematode

migration

may

also

be

seen,
10

as

illustrated

in

the

present

case

(Fig
.

2
)
.

G i v e n

t h e

p r o t e a n

m a n i f e s t a t i o n s

and

frequent

overlap

of

pulmonary

tuberculosis

in

areas

of

the

world

where

paragonimiasis

is

common,

tuberculosis

is

initially

suspected


Figure 1: Computed
tomography of the chest illustrating a large
cavitary infiltrate in the right lower lobe.

CONCLUSIONS

Although

an

ELISA

is

available

through

the

Centers

for

Disease

Control

and

Prevention,
14

confirmation

of

disease

by

visualization

of

ova

or

adult

worms

is

preferential
.

Adult

worms

are

infrequently

identified

in

patient

specimens
;

however,

if

present,

the

adults

are

distinctively

large

and

contain

a

conspicuous

cuticular

spine
.
1

For

lung

parenchymal

involvement,

acid
-
fast

stains
15

and

standard

ova

and

parasite

examinations

of

sputum

may

reveal

diagnostic

ova,

but

these

techniques

are

insensitive
.

Speci mens

procured

by

bronchoscopy

with

biopsy

or

other

means

are

more

sensitive

and

may

reveal

large

parasite

ova

(
60
-
120
µm)
1

in

addition

to

eosinophilic

infiltration

and

possibly

granulomas

in

biopsy

specimens
.
16


Ova

may

be

particularly

difficult

to

locate

in

the

event

of

a

pleural

effusion

with

no

parenchymal

involvement
.

In

such

cases,

a

careful

epidemiologic

history

and

serology

are

most

useful,

as

pleural

fluid

eosinophilia

is

an

inconstant

and

non
-
specific

finding
.
6

Serol ogy

may

al so

be

of

use

in

other

instances

in

which

pathologic

samples

are

nondiagnostic

or

when

sensitive

areas,

such

as

the

eye

or

b r a i n s t e m,

w o u l d

have

to

be

biopsied

to

attain

a

pathologic

diagnosis
.
Ova

from

Paragonimus

species

can

be

discerned

from

related

genera

such

as

Schistosoma

by

birefringence

under

polarized

light,

as

in

the

current

case

(Fig
.

3
B),

and

the

presence

of

an

operculum
.

Cytologic
,

in

contrast

to

histologic,

analysis

may

be

most

beneficial

in

equivocal

cases,

as

ova

morphology

is

less

likely

to

be

compromised
.
1




The

best

studied

antihelminthics

for

this

disease

are

praziquantel
18

and

triclabendazole
.
19

Tr i c l a b e n d a z o l e

is

not

available

in

the

United

States
;

therefore,

the

recommended

regimen

in

the

Unites

States

is

praziquantel

given

as

25

mg/kg

orally

three

times

per

day

for

two

days
.