paraplegia due to aspergillosis - Bone & Joint

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HAl. 3UJ. England. C. Jones. FRCS. Senior. Orthopaedic. Registrar ..... Erickson. DL, Chou. SN,. Seljeskog. EL. A.spergi//us infections of the lumbar disc spaces:.
PARAPLEGIA SUCCESSFUL

DUE

TO

CONSERVATIVE BARRY

From

ASPERGILLOSIS

TREATMENT

FERRIS.

Westminster

OF

CLEDWYN

Children’s

TWO

CASES

JONES

Hospital,

London

Aspergilus infection of the spine is rare; for it to lead to paraplegia is still more rare. When this does it is usually treated by decompression and antifungal agents, but the results have usually been poor. We report two cases of successful conservative treatment of Aspergilus paraplegia in patients with chronic

occur

granulomatous

Infection in man.

disease.

by fungi The usual

of the genus site ofinfection

is uncommon is the lungs but spread

to other organs can occur and there have been reports intervertebral disc infection, vertebral osteomyelitis extradural

abscess.

There

tion and multiple

Aspergillus

have

been

very

few

of and

reports

of

a bilateral discharging

extensor sinuses

three months foot drop.

report responded

a number of further mal neurology; his

infections, vertebral

were

quiescent

3).

Case

2. An

cases of to antifungal

paraplegia

Aspergil!us

treatment.

CASE REPORTS Case 1. A six-year-old boy was referred minster Children’s Hospital for a bone plant. months,

He

was an adopted had developed

bone after a trivial multiple infections, drainage.

until,

which

to the marrow

child who, osteomyelitis

at the age of six of the occipital

graze. Thereafter he requiring antibiotics

at the age

Westtrans-

of I 5 months,

suffered from and surgical

chronic

granulo-

matous disease was diagnosed. A further series of infections followed and at the age of five years he developed a right-sided empyema with a subphrenic extension. This was aspirated and Aspergil!usfumigatus was cultured. He was

treated

with

clotrimazole

to eradicate osteomyelitis

the infection of the upper

nosed

I and

(Figs

2). He

and

this

failed

and at the age of seven thoracic vertebrae was

years diag-

received

plant with amphotericin transplant was unsuccessful. At the he was

age

found

of eight

B. Ferris. Northwick HAl 3UJ.

FRCS. Wellcome Park Hospital. England.

Research Watford

C. Jones. Westminster England.

FRCS.

Requests

for reprints

1985 British 0301 620X85 (

800

a bone

marrow cover,

he presented paraplegia

with with

transbut

the

incontinence;

sparing

Fellow Road,

Harrow,

of sensa-

Middlesex

be sent

Editorial Society 5171 S2.00

to Mr

of Bone

London

B. Ferris. and

Joint

Surgery

SW1P

later,

At the latest

infections sented and

an

the review

(Fig. adopted

since with

abscess

only

residual

when

He he

had was

aged

was

14 he had

a

had

but there was no abnorinfection and his sinuses boy

suffered

At the

of the

abnormality

he was

Italian

infancy.

pneumonia

abscess was lusfiit;zigatu.s-

age

ofthe

upper

right

side

from

of five

recurrent

years

he pre-

lobe

ofhis

right

lung

of his

chest

wall.

The

drained and culture of the pus grew Aspergiland Aspergi/lu.siiigei’. He was treated with

amphotericin B and 5-fluorocytosine. Further investigations revealed that he had chronic granulomatous disease and. after granulocyte transfusion, he was referred for a bone marrow had paraplegia. with sparing

transplant. On admission he of sensation and sphincter

control,

extensor

and

Radiographs vertebrae extradural

a

bilateral

showed collapse (Figs 4 and 5) and block

intravenous daily) for

(Figs

amphotericin three months

plantar

of several a myelogram

5 and

6).

He

B at maximum and then with

response.

upper thoracic revealed an

was

treated

with

doses ( 1 mg/kg oral miconazole

(1 g daily). The weakness improved after eight days and by the fourth month he was able to walk. He was fitted with a brace and by the seventh month had regained full function; the only remaining abnormality was a bilateral extensor plantar Four years ology

Orthopaedic Registrar Dean Ryle Street, should

but

antibiotic

to have

Senior Hospital,

emetine,

response. hack and

almost moribund. He was treated with intravenous amphotericin (0.25 mg/kg bodyweight daily rising to I mg/kg daily) and the paraplegia slowly improved until.

paraplegia secondary to Aspergi!!us infection and, in a search of the literature, we have found no report of successful conservative treatment of this condition. We two

plantar on his

(Fig.

response. later he was

well

with

no abnormal

neur-

7).

2AP.

DISCUSSION Infection

by A.s’peigi//u.s-

becomes although

invasive in the presence of debilitating there are reports of patients in whom THE

is rare.

JOURNAL

The

OF

BONE

fungus

AND

usually

JOINT

only

disease. there was SURGERY

PARAPLEGIA

Fig.

DUE

I

Fig.

Fig. (‘ase I . Figure vertebrae. The Figure 2-Lateral and lytic areas:

VOl..

67 B. No.

5. NOVEMBER

1985

801

TO ASPERGILLOSIS

3

1 -This patient had destruction of several upper thoracic second and third right ribs show lytic areas and are expanded. tomogram of upper thoracic vertebrae showing sclerosis note the absence ofsequestrum formation. Figure 3-Radiograph eight years after presentation.

2

802

B. FERRIS,

Fig.

4

C. JONES

Fig.

5

Case 2. Figure 4-Radiograph showing vertebrae. Figures 5 and 6-Myelogram from the fifth thoracic vertebra (Fig. 5) (Fig. 6). Figure 7-Radiograph four

Fig.

destruction of upper showing an extradural to the sixth cervical years after presentation.

THE

6

thoracic block vertebra

JOURNAL

OF

BONE

AND

JOINT

SURGERY

PARAPLEGIA

no underlying

cause

(Grossman

and Lerner 1977). Many described as pathogenic, A.

tiaras.

1974:

A.

nidulans

Grossman of these

I 977);

1975) A

and

A.

. flimigatus

Predisposing surgery and

factors radiotherapy.

link

infection

between

1975;

Seligsohn,

species ofAspergi!lus such as A. fumigatus, (Bujak, Kwon-Chung is

Rippon

the

use

et

neoplasia, reports of

a!.

cardiac imply a

oral

803

TO ASPERGILLOSIS

one died (Seres, Ono and Benner Dietz ci a!. 1982; Chee and Poh

have been A. niger, and Chusid

terreus (Seligsohn the commonest.

include Several and

DUE

contracep-

paraplegia from prognosis. In children, infection (Bujak

of ci a!.

an

epidural

of

five

granulomatous

X-linked

deficiency

abscess

reported

the spine 1974; Tack

chronic

1972; Byrd 1983). Clearly.

without

cases

disease. of the

This

enzyme

enzyme needed for destroying nisms such as Staphylococcus

also are at risk (Byrd. Weiner and McGee 1982), as are diabetics (Mawk et a!. 1983). In addition, there have been reports of infection during pregnancy (Dietz et a!. 1982) and in patients with chronic granulomatous

Several antifungal agents are ing amphotericin B, 5-fluorocytosine. conazole and miconazole these, such as amphotericin

disease

and

hepatic

ment

continues. In conclusion,

ing

(Bujak et a!. 1974; The organism usually pneumonia, although

Tack et a!. 1982). gains entry via the lung, causit may lie dormant only to

become active later, as reported in a patient who developed paraplegia during pregnancy (Dietz et a!. 1982). In the presence of pneumonia. dissemination to the heart, kidney and tion rarely

gut is said spreads

Haematogenous

to occur to bone

spread

occurs

spread to bone is usually (Grossman 1975; Tack two

cases.

sinus

Clinically formation

though dense

new

bone

difficulty. small

Reviewing the literature A.s-pergil/us paraplegia which spine

in adults

tenor

decompression

a!.

lytic

lesions

1983).

paraplegia

VOL.

67 B. No.

Four which

patients did

5. NOVEMBER

1985

despite

believe

but

with

and

is

report of treated by

of the pos-

Six were

abscesses treatment,

all

and

be

recessive an orgaspp.

available, clotrimazole,

includketo-

whilst

Aspergi!lus

Dr

Terrett

treatis a rare

mortality.

poor.

but

antifungal

The

in children

agents

K.

Hugh-Jones

for

typing

iS, Kwon-Chung Ki, Chusid Mi. in a boy with chronic granulomatous by a mutant strain of Aspergi//u.s1974:61 :361-7.

Byrd

BF Ill, Weiner MH, McGee abscess.JAMA l982;248:3l38

Chee

YC,

Poh

obstructive

Dietz

R, Huber osteoclasis

Grossman Mawk

pri-

had

a

died had

formation.

now

that to

and

Linda

is

a significant

with

P. Aichroth

cases,

abscess

Bujak

features

infection treated

agents.

epidural

improve

Mr

these

four children

for

the

can

be

permission

to

manuscript.

REFERENCES

a notable

1979),

we could find no was successfully

we

treatment

thank

Aspergi!!us

to be monitored

appears

SC. Aspergillus airway disease.

Convent L, Van de Meirop sis. Acta Orthop Be/g

antifungal

with not

conservative successful.

poor

(Convent ci a!. 1979). Some of B, are toxic, so that renal

need

in adults

report

manly disc infections; four recovered (one of whom had a paraplegia which resolved) and two died (Grossman 1975; Seligsohn ci a!. 1977; Convent ci a!. 1979; Mawk ci al.

function

of

peroxide-producing aureus and Aspergi!lus and

the lung in our

there

and

c/ a!.

in granuloma

of paraplegia,

outlook

We

in children

Radiologically

Ten cases of Aspergi!!us have been reported. all and

infec1977).

results

cause

infection of bone results in the fungus may be cultured,

absence of sequestration (Convent which were present in our two cases.

conservatively.

but

by direct invasion from 1982) as happened

with with

in adults,

but

ci a!.

Aspergillu.s’ from which

sometimes

in 25% ofcases, (Seligsohn et

This

a

myeloperoxidase,

addictive drugs and intravenous catheterisation (Seligsohn et a!. 1977; Convent, Van de Meirop and Blijweert 1979). Patients on immunosuppressive therapy tives.

carries

paraplegia 1982). These

ci al.

a!. 1982; in adults

ci

G, and

M. Aspergillosis

JL, Ono compression:

Tack KJ, Rhame litis: report 1982;73:295

H,

epidural Po.stgrad

Aspergillus

spinal

epidural

abscess in a patient MedJ 1983:59:43 5. ofvertebral

BrJ

Radio/

1975:48:57

with

aspergillo-

Volkmer I. Aspergillosis of the lung Neuroradio/ogv I 982 : 23 : 2 19 2 1.

ofbone.

DL, disc

Seligsohn R, Rippon Arch Intern Med

Seres

0,

paraplegia.

Erickson of the lumbar 58:270-4.

ZA. 9.

L, Blijweert D. A case 1979:45: l4l 50.

Thetter

JR,

Osteomyelitis and pneumonia disease ofchildhood caused nidu/an.s-. Am J C/in Pat/w/

with

9.

Chou SN, Seljeskog EL. A.spergi//us infections spaces: report of 3 cases. J Neuro.s-urg 1983:

Lerner

JW,

l977:

SA. Aspergillus 137:9 I8 20.

Benner EJ. Aspergillosis case report. J Neurosurg

FS, Brown B, Thompson of four cases and review --300.

terreus

presenting 1972,36:221 Jr. Aspergillus of the literature.

RC

osteomyelitis. as spinal 3.

cord

osteomyeAm J Med