Primary

Coccidioidomycosis Massive

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LEE

in Children Pleural

PINCKNEY1

AND

R. PARKER’

before admission. Three days after the trip she, her sister, and three family dogs developed flulike syndromes and were treated with antibiotics. A week later she developed fever, neck and chest

pain,

lingular

and

initially proved

and therapy

radiograph

1 week later showed

positive.

were

dogs

to have

complement

had

fluid

been

sent

of California, fixation

sis,

Her

(fig.

2).

chest

at Stanford

A

effusion.

without

cells

to the

School

Davis, where

titers

radiograph

Subsequent

for presumed

showed

to

of

they

of 1 :16 and were

left anterior

with

amphotericin

ment

fixation

was

stopped

B for titers

1 month

were

when

she

pleural hilar

effusion lymphade-

lymphadenopathy, Thoracentesis cells/mm3. and

1 :4 and

left

bilateral

mediastinal

improved

reached

developed

to

coccidioidomyco-

a large

thoracentesis,

lower lobe infiltrate were seen. fluid with eight polymorphonuclear

and

left

yielded clear She was treated rapidly.

a high

Compie-

of 1 :8. Therapy

granulocytopenia,

but

she

asymptomatic.

Reports Case 3

This 2-year-old Mexican-American male accompanied his family on a trip into the San Joaquin Valley 2 weeks before the onset of his illness. He presented with fever, tachypnea, and

,

was identified. Thoracentesis yielded clear straw-colored fluid, negative on culture. She failed to respond to penicillin and cephalexin and, after 1 week, was transferred to Stanford on admission

demonstrated

a large

February

1978

right fluid.

pleural Protein

blood

by

his

improve

hospital.

local

with

physician

a 10 day

A chest

with

course

radiograph

oral

and

was

revealed

a

effusion which, at thoracentesis, yielded was 5.2 g/100 ml; glucose, 76 mg/100 ml;

cell count,

and six negative,

1 440/mm3

polymorphonuclear and microscopic

(85 lymphocytes,

nine

cells). Bacterial culexamination of the

to improve. After several weeks he was transferred to Children’s Hospital at Sanford where a chest radiograph confirmed the large right pleural effusion and showed right middle and lower

lobe infiltrates The patient ioidomycosis

(fig. 3). was scheduled complement

ously was discovered

for pleural fixation

titer

to be positive

biopsy

when

drawn

3 weeks

a coccidprevi-

at a 1:16 dilution.

Another

titer was immediately drawn and found positive at 1:256. Fungal cultures of the pleural fluid taken 3 weeks previously grew Coccidioides immitis. Amphotericin B therapy was begun 30

days after the patient’s first visit to a physician and was continued 14 weeks until the complement fixation titer had decreased to 1:16. The pleural effusion and pulmonary infiltrates gradually

Received July 15, 1977; accepted after revision October 17, 1977. ‘Department of Radiology, children’s Hospital at Stanford and Stanford requests to B. R. Parker. Ray Society

treated to

stained fluid revealed no organisms. A coccidioidomycosis skin test was positive. He was treated with erythromycin but failed

2

130247-249,

was failed

to a local

monocytes, tures were

The patient, a 6-year-old white female, had been on a camping trip with her family through the San Joaquin Valley 6 weeks

Roentgen

He

and white

right-

coccidioidomycosis, histoplasmosis, blastomycosis, and mumps were all negative. A pleural biopsy at open thoractomy revealed caseating granulomas and fungal forms characteristic of coccidioidomycosis. Acid-fast stains were negative. The complement fixation titer at the time of pleural biopsy was 1:32 and later reached a level of 1:128. She was treated with amphotericin B for 11 weeks and has had no evidence of recrudescence. Follow-up radiographs show a stable right upper lobe nodular infiltrate and right costophrenic angle blunting.

Am J Roentgenol © 1978 American

and

ampicillin.

large clear

Hospital. radiograph

weakness,

admitted

sided pleural effusion and right hilar lymphadenopathy (fig. 1). Another thoracentesis yielded fluid with a protein of 8.3 g/100 ml, 9,200 white blood cells/mm3 (61% polymorphonuclear, 39% mononuclear), and negative culture. Skin tests for tuberculosis,

Case

to erythromycin.

a large pleural

straw-colored

University

Hospital

nopathy,

elusive,

This 10-year-old white female had moved to the San Joaquin Valley of California with her family 6 months before the onset of her illness. Two weeks before admission she developed fever and tachypnea for which she was treated with antibiotics. One week later she was hospitalized and a right pleural effusion

A chest

family

a

Her symp-

A skin test for coccidioidomycosis

Medicine,

found

Children’s

1

University

The

was changed

clear

culture.

demonstrated

on ampicillin.

started on therapy for disseminated coccidioidomycosis. Five weeks after the onset of symptoms, she was admitted

remained Case

yielded

and with a negative

largely because the presenting feature in each case was a large pleural effusion. This presentation is unusual but well documented in adults; it has not previously been reported in children.

Case

radiograph

progressed,

A thoracentesis

Coccidioidomycosis is an endemic disease in the southwestern United States, but can be seen elsewhere in the country as travel to this area becomes more common. The clinical and radiographic manifestations are variable and occasionally the diagnosis is difficult. We have recently encountered three children with coccidioido-

A chest

and she was started

chest

Veterinary

the diagnosis

dyspnea.

infiltrate,

toms

was

in whom

with

Eftusion

BRUCE

Large pleural effusion as the presenting feature of coccidloidomycosis in childhood has not previously been reported and may cause significant diagnostic difficulty. Three children are described who required prolonged hospitalization and therapy with amphotericin B. Primary coccidioldomycosis should be included in the differential diagnosis of large pleural effusions in pediatric patients who have been in the endemic area. Such effuslons suggest a severe form of the disease and may represent evidence of acute dissemination.

mycosis

Presenting

University

247

School

of Medicine,

Stanford,

California

0361 -803X/78/0200

94305. Address

-0247

reprint

$02.00

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248

PINCKNEY

AND

PARKER

,

-a

I_

Fig. 2.-Case 2. Admission chest radiograph demonstrating large pleural effusion. Left lower lobe infiltrate is obscured by fluid. Subsequent films also showed hilar and mediastinal lymphadenopathy.

TBJ#{243} Fig. 1 . -Case 1 . A, Admission right-sided pleural effusion. B,

lymphadenopathy enchymal infiltrate.

resolved.

Minimal

end of therapy

chest

radiograph

Chest tomogram and large right loculated pleural

costophrenic

angle

had disappeared

1 year

demTrating

large

showing right hilar effusion without par-

blunting

present

at the

later.

Discussion

The full spectrum tations of primary mented

and

will

of clinical and radiographic coccidioidomycosis is not

ponent, the small adult and pediatric pulmonary infiltrate ing of a costophrenic region

of

the

be discussed

here

well

[1-4].

manifesdocu-

One

com-

pleural effusion, is reported in both cases, usually in association with and usually appearing only as bluntangle or pleural thickening in the

infiltrate.

In

one

series

of

18

pediatric

[5], four had minimal pleural effusion, an incidence approximating the reported rate of 20% in adults [3]. Another series of 120 children disclosed only eight patients

with

pleural

effusions,

but

many

patients

in this

Fig. 3.-Case 3. Admission chest radiograph demonstrating right pleural effusion, right hilar lymphadenopathy, and right infiltrate.

large

basilar

series

had chronic disease [6]. Massive pleural effusion in adults is clearly documented in several early series [3, 6] but has not been described previously in children. The youngest reported case we could find occurred in a 19-year-old patient reported by Ophuls and Moffitt [7] in 1900. Interestingly, that is the first recognizable report of acute primary coccidioidomycosis [8].

Most have

children

symptoms,

with and

primary most

coccidioidomycosis

symptomatic

patients

never recover

within 10 days without therapy [5]. The three cases with massive pleural effusion presented here were clearly more serious than the vast majority, including those with small pleural effusions. In two of the three cases dissemination was documented by serologic criteria

COCCIDIOIDOMYCOSIS (complement fixation titers three required prolonged

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with

amphotericin

B.

In

of greater than hospitalization children,

the

WITH

MASSIVE

PLEURAL

REFERENCES

1 :16), and all and therapy presence

of

a

massive pleural effusion may be a harbinger of dissemination similar in significance to paratracheal lymphadenopathy [9]. In all three of our cases diagnosis was delayed for 1 month or more as each patient was treated for bacterial pneumonia. Two of the three cases were referred without diagnosis to our center from hospitals which see numerous cases of coccidioidomycosis each year. The diagnosis in one case was not suspected prior to thoracotomy. Even though physicians of the endemic area frequently see coccidioidomycosis, the unusual presentation of the three children reported here caused significant diagnostic difficulty. Primary coccidioidomycosis must be considered in the differential diagnosis of large pleural effusions in pediatric patients who have been in the endemic region. Such effusions suggest a severe form of the disease and may represent evidence of acute dissemination.

249

EFFUSION

1 . Fraser AG, Pare JAP: Diagnosis Philadelphia, Saunders, 1970

2. Greendyke

WH, Resnick

of Diseases

DL, Harvey

gen manifestations of primary Roentgenol 109:491-499, 1970

3. Jamison HW, Ray AC: Roentgen domycosis. Radiology 48:323-332,

of the Chest.

WC: The varied

coccidioidomycosis.

findings

roentAm

J

in early coccidioi-

1947

4. Casteilino RA, Blank NB: Pulmonary coccidiomycosis: the wide spectrum of roentgenographic manifestations. Calif Med 109:41-49, 1968 5. Richardson HB, Anderson JA, McKay BM: Acute pulmonary coccidioidomycosis in children.J Pediatr 70:376-382, 1967 6. Birsner JW: Roentgen aspects of 500 cases of pulmonary coccidioidomycosis. Am J Roentgenol 72:556-573, 1954

7. Ophuls W, Moffitt HC: A new pathogenic mould (formerly described as a protozoon: Coccidioides immitis pyogenes). Philadelphia MedJ 5:1471-1472, 1900 8. Fiese MJ: Coccidioidomycosis. Springfield, Ill.,Thomas, 1958 9. CoIwell JA, Tillman SP: Early recognition and therapy of disseminating coccidioidomycosis. Am J Med 31:676-691, 1961