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i 035

Case Report “



:.:

‘.



Mucormycosis of the Cuboid R. Chaudhuri,1

B. McKeown,1

Mucormycosis is infection known to pecially those with diseases, and renal

ebral,

Osteomyelitis Causing Avascular Bone: MR Imaging Findings

although

D. Harrington,2

R. J. Hay,3 J. B. Bingham,1

an uncommon but highly invasive fungal affect immunocompromised patients, esdiabetes mellitus, hematologic malignant failure. The most usual form is rhinocer-

it can have

abscess

foot

the

intestinal, disseminated, or cutaneous manifestations [i ]. We report a case of a renal transplantation patient who had pain and swelling in

diately

remote

of

fungal

of bowel

monas thema

perforation sepsis.

due

While

developed

he was

track

to cytomegalovirus

he

was

in the lateral

hospitalized,

aspect

Received March 1 6, 1 992; accepted 1 Department of Radiology, Division

admitted

for treatment

infection

and

pain,

swelling,

of his right foot.

after revision of Radiological

from

the

cuboid

tration

of

hyperemic

short

AJR 159:1035-1037,

November

1992 0361-803X/92/1595-1

and raised fungal

abscess

lesions

a clinical

growth

was

imme-

showed

no

suspicion

obtained

on

were normal apart from and a lucency corresponding to a

to the skin.

patient

was

No bone

referred

for

abnormality

was

scintigraphy

and

The radionuclide bone scan, (MOP), showed intense radiobone on equilibrium and static

showed

low

signal

on the

Ti -weighted

soft

tissues

dimeglumine (Fig.

1 0).

(0.1 Signal

mmol/kg)

intensity

except was

(500/

affecting only after adminishigh

in the on

the

(STIR) image (i 800/30/i 50 [TRITE/TI]) and in the surrounding soft tissues (Fig.

1 E).

Surgical

exploration

and biopsy

of the cuboid

bone

were

under-

taken. The subcutaneous tissues were infarcted with pus formation. Pseudomonas was cultured from the soft tissues. Results of the bone biopsy showed extensive necrosis of trabeculae and the marrow

ery-

St. Thomas

SE1 9AT,

St. Thomas

035 0 American

bone The

TI inversion recovery in the affected bone

both

Hospital,

St., London

three

of the foot

thickening

gadopentetate

St., London

of Trauma

Hospital,

a further

last

with a surrounding rim of lower intensity, bone (Fig. 1C). No enhancement occurred

of Dermatology,

Guy’s

no

and

uptake (Fig.

Department

St. Thomas

The

image

Department

Surgery,

bone,

inflammation

although

i A).

images

4

Hospital,

of Pseudomo-

1 B). MR

3

Guy’s

a culture

phases, with a large central area devoid of radionuclide

of Renal Medicine, Guy’s Hospital, St. Thomas St., London SE1 9RT, United Kingdom. and Orthopaedic

cuboid malleolus.

the cuboid (Fig.

20 [TRITE])

A soft-tissue

May 18, 1992. Sciences, Guy’s

the

lateral

soft-tissue

Department

2

yielding

MR imaging to exclude osteomyelitis. using “Tc-methylene diphosphonate nuclide uptake surrounding the cuboid

Pseudoand

site,

on plain radiographs

extensive

.

transplantation,

from the

infection,

Findings

causing

A 63-year-old man with polycystic kidney disease was treated with continuous ambulatory peritoneal dialysis for 8 months before receiving his first cadaveric renal transplant in June i 991 The onset of graft function was delayed for 6 weeks because of acute tubular necrosis and mild rejection. Initial immunosuppression was with prednisolone, azathioprine, and antilymphocyte globulin, which was given for i 0 days. Cyclosporin was started after the course of antilymphocyte globulin had been completed. No further episodes of rejection occurred, and his plasma creatinine level fell to about 200 mol/l. He was never neutropenic. after

this

culture.

Case Report

months

from

of surrounding

discernible

Three

drained

above

evidence

osteomyelitis To our knowl-

edge, this is the first description of mucormycosis osteomyelitis and avascular necrosis.

and J. D. Spencer

nas. We noted subsequent development of two fistulous tracks from the cuboid bone to the skin, which did not communicate with each other, as well as two other superficial abscesses on the dorsum of

pulmonary,

the foot and was found to have mucormycosis causing avascular necrosis of the cuboid bone.

was

Necrosis

United

United

Kingdom.

St., London

Roentgen

SE1 9AT,

SE1 9AT,

Ray Society

United

Kingdom.

Kingdom.

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i 036

CHAUDHURI

ET AL.

AJR:159,

November

1992

Fig. 1.-A, Plain radiograph of right foot shows normal appearance of cuboid bone with soft-tissue corresponding to track (arrow). B, Scintigram of lateral aspect of right foot shows central area of no radionuclide uptake (arrow) over bone, surrounded by a rim of high radionuclide uptake. C, Sagittal Ti-weighted MR Image of right foot shows low signal within cuboid bone with a rim of lower intensity (arrow). D, Axial contrast-enhanced MR Image through cuboid bone shows no enhancement of bone. Track to

defect cuboid

signal skin is

well visualized. STIR MR image shows high signal In cuboid bone (arrow)

E, Coronal

space with a predominantly acute inflammatory cellular infiltrate and numerous broad aseptate fungal hyphae having the typical morphologic appearance of mucormycosis. A zygomycete fungus, Rhizopus rhizopodoformis, was subsequently grown on culture. The cuboid bone was excised, the wound was debrided, and IV amphotericin was administered for 4 weeks. The patient mad a good recovery and was

well with

discharged

a functioning renal transplant.

soft tissues

in right foot.

was an inpatient on the same ward as our patient. It is, however, surprising that, if infection had been transmitted by inhalation,

no rhinocerebral

disease

or evidence

of dissemi-

nated mucormycosis infection occurred other than in the foot of our patient. It remains possible that secondary infection occurred through the initial abscess drainage site from airborne spores, but the fungus was never cultured from this site. This fungus causes tissue ischemia and

Discussion

and adjacent

invasion of blood vessels, leading hemorrhagic infarction depending

to on

Mucormycosis is a fungus of the class Zygomycetes, family Mucoraceae, and usually of the genera Rhizopus, Rhizomucot, orAbsidia. These fungi are ubiquitous in the environment, and, it has been said, in hospital wards, but environmental cultures taken regularly throughout a 10-year period in another institution [2] have seldom yielded the organism found in this case, Rhizopus rhizopodoformis. Patients particularly at risk for infection with this agent are those who are diabetic

arterial or venous involvement, and an intense inflammatory response. Isolated cuboid bone involvement with total avascular necrosis suggests loss of periosteal and nutrient arterial supply or diffuse central venous occlusion. As it spreads through perivascular spaces, bone destruction is an uncommon and late finding in rhinocerebral disease even when the

or immunocompromised,

ings. This patient was referred for scintigraphy and MR imaging because the clinical presentation was of cellulitis, and under-

roids,

hyperglycemia,

tation

patients),

whether

from

antirejection

or neutropenia

high

therapy [3].

The

doses

of ste-

(as in transplaninfection

has been

surrounding

are reflected

tissues

are extensively

involved.

in the radiographic,

These

scintigraphic,

features

and MR find-

reported in patients undergoing hemodialysis who were receiving desfemoxamine for iron overload [4]. Transmission is usually by inhalation or by direct invasion of the bowel in

lying osteomyelitis radionuclide

activity

malnutrition. Skin and subcutaneous tissues subjected thermal burns and nosocomial infections have become

appearance

in this case was not that of osteomyelitis,

fected screening

after application of dressing

of contaminated packs

in this

dressings case

to in-

[2, 5], but

did not show

any

evidence of mucormycosis infection. It is of particular interest that another patient with renal failure who had extensive rhinocerebral mucormycosis, diagnosed 3 months previously,

scintigraphy

would

that of avascular

was suspected. In osteomyelitis, bone be expected to show a focal increase in in the affected necrosis.

The

bone.

normal

The

scintigraphic

or reactive

bone

but ad-

jacent to the ischemic region showed increased radionuclide uptake in contrast to the lack of uptake in the central avascular portion of bone. This is compatible with the normal radiographic findings in early avascular necrosis as the mineral content of the bone is preserved while the cellular elements

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AJA:i59,

November 1992

MR OF MUCORMYCOSIS

in the marrow are affected by vascular occlusion [6], although with such marked scintigraphic findings, it is surprising that no evidence of osteoporosis was seen. MR imaging is said to be more sensitive and specific than mTc-MDP bone scintigraphy [7] in distinguishing between soft-tissue cellulitis, abscess, osteomyelitis, and avascular necrosis because of its increased spatial resolution. Low signal on the Ti-weighted image affected only the cuboid bone, suggesting a decrease in marrow fat and an increase in water content, with no enhancement after administration of gadopentetate dimeglumine except in the hyperemic soft tissues. The high signal intensity on the STIR image both in the affected bone and in the soft tissues reflects a high water content, as occurs with hyperemia, edema, or inflammation, and would be compatible with osteomyelitis and noninfectious inflammatory conditions as well as early avascular necrosis [7, 8]. However, a rim of low signal intensity also was present around the cuboid bone on the Ti-weighted image, which is consistent with class C avascular necrosis as described by Mitchell et al. [8] and does not occur in osteomyelitis unless it is long-standing. The different patterns described reflect the evolution of osteonecrosis, of which osteomyelitis can be the

cause, and depend

on the signal changes

due to the amount

of marrow fat, blood, water, and fibrosis present; other than with class C avascular necrosis, osteomyelitis is usually easily distinguished from avascular necrosis.

OSTEOMYELITIS

1037

AND AVN

Mucormycosis has previously been associated with a high mortality rate. In this case, the scintigraphic and MR imaging studies were critical in the diagnosis of avascular necrosis. Owing to the rapid diagnosis, appropriate surgery and therapy could be offered to our patient. Nonetheless, the excellent and rapid response to treatment in this case is unusual in this disease. REFERENCES 1 . Hammer recipient.

GS, Bottone EJ, Hirschman SZ. Mucormycosis Am J Clin Pathol 1975;64:389-398

2. Gartenberg G, Bottone mucormycosis (Rhizopus

EJ, Keusch GT, Weitzman

in a transplant

I. Hospital-acquired

rhizopodiformis) of skin and subcutaneous tismycology and treatment. N EngI J Med i978;299:

sue: epidemiology, 1115-1118 3. Anderson RJ, Schafer LA, Olin OB, Eickhoff TC. Infectious risk factors in the immunosuppressed host. Am J Med 1973;54:453-460 4, Boelaert JA, Fenves AZ, Cobum JW. Mucormycosis among patients on dialysis (letter). N EngI J Med i989;321 :190-1 91 5. Hammond DE, Winkelmann AK. Cutaneous phycomycosis: report of three cases with identification ofRhizopus. Arch Dermatol 1979;1 15:990-992

6. Greyson NO. Radionuclide eds. Diagnostic

imaging,

1535-1547 7. Unger E, Moldofsky osteomyelitis

bone scanning. 1st ed. New

P, Gatenby

by MR imaging.

AiR

York:

In: Grainger Churchill

A, Hartz W, Broder

AG, Allison OJ,

Livingstone,

1986:

G. Diagnosis

of

1988;150:605-610

8. Mitchell DG, Aao VM, Dalinka MK, et al. Femoral head avascular necrosis: correlation of MR imaging, radiographic staging, radionuclide imaging, and clinical findings. Radiology 1987;162:709-715

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i 038

Case

Rapid Growth

of Femoral

J. P. Crabbe,1

W. Martel,1

Herniation

pit of the femoral

Herniation

fewer

proved

neck

herniation

than i4 months.

important

is a common,

pit more

In order

to recognize

that

in size to i 7 X 14 mm and had become

benign

than

doubled

to avoid misdiagnosis,

such

rapid

growth

in

it is

of this benign

lesion is possible. Case Report A

55-year-old was

woman who had had pain in the left hip for several referred

to the

Section

of Orthopaedic

Surgery

for

evaluation. The clinical diagnosis at that time was left-sided ischial bursitis, and findings on the initial radiographs and all subsequent radiologic studies of the left hip were normal. However, incidentally noted

on the initial

radiographs

of the pelvis

and hips

was

a well-

defined, 9 x 10 mm lucent lesion, with sclerotic margins, in the right femoral neck (Fig. 1A). The patient had not had any signs or symptoms related to the right hip. At this time,

the diagnosis

made, but osteoid osteoma Two

months

of a probable

was considered

after the initial plain films,

herniation

pit was

up for a further

radiograph

At that time,

bone scan was

obtained (Fig. 1D). This showed normal flow and blood-pool phases, but delayed images revealed a focal region of abnormally increased uptake of radionuclide within the right femoral neck, corresponding to the lesion seen on radiographs. The lesion was thought to be benign, most likely a hemiation pit, but it was decided to do follow-up studies. A radiograph obtained 8 months after initial presentation showed that the lesion had increased Received

AJR

April

1

Department

2

Section

1 5, 1992;

of Radiology,

of Orthopaedic

159:1038-1040,

accepted

November

after revision

University

of Michigan

1992 0361 -803X/92/l

after

initial

Throughout relative

this

to the

time

right

hip.

the The

patient lesion

had

was

still

remained thought

asymptomatic

with a herniation pit, but because of the rapid increase in size, CT scans and MR images were obtained (Figs. 1E-1 H). CT scans showed a well-defined lesion of soft-tissue attenuation, with sclerotic margins, on the anterior surface of the femoral neck. The lesion had a cortical roof and a focal perforation 1 mm in diameter. MA images

showed a lesion that had uniformly low signal on Ti -weighted images and no significant enhancement after administration of contrast material. On T2-weighted intensity. this

time,

the

images, imaging

the lesion was of uniformly characteristics

were

high signal

considered

typical

for an enlarging, but otherwise unremarkable, hemiation pit. Despite the benign prognosis, the patient decided she wanted a biopsy of the lesion; this was done 14 months after her initial presentation. The lesion was resected by using a CORB biopsy needle (Zimmer Inc. , Warsaw, IN) and a pituitary rongeur. Macroscopically, the lesion

consisted of “slimy gray-white tissue.” Histologic examination showed a dense fibrous connective tissue with poorly defined foci, which were of looser texture and myxoid appearance. Fragments of cartiIage, bone, and fibrinous material also were present.

Discussion

A herniation “reaction

area,”

pit of the femoral

neck arises in relation to the

an area of sclerotic

bone,

covered

by collag-

enous tissue and neocartilage, present in the anterolateral region of the upper femoral neck in most adults [2]. lngrowth of these fibrous and cartilaginous elements occurs through a perforation

in the

subcortical

location

1500 E. Medical C American

pres-

to be consistent

cortex; to form

these the

tissues

then

radiographically

expand

Roentgen

Center

Or., Ann Arbor,

Ray Society

Ml 48109.

in a

identifiable

1500 E. Medical Center Or., Ann Arbor, Ml 48109. Address reprint requests to W. Martel.

Hospitals,

595-1038

14 months

(Fig. 1 B).

and another

i C).

May 1 5, 1992.

University of Michigan Hospitals,

Surgery,

was obtained.

lobulated

6 months,

entation, the lesion had increased in size to 23 x 16 mm; thin sclerotic margins were still present, but the contour was more lobulated (Fig.

as a less likely possibility.

a triple-phase

slightly

The lesion was followed

At

months

Pit

and L. S. Matthews2

entity that was first formally characterized by Pitt et al. in i 982 [1 1. The lesion is seen as a small rounded radiolucency, with a thin sclerotic margin, situated on the anterior surface of the femoral neck. In their original paper, Pitt et al. described two cases in which herniation pits increased in size, one during a 6-year period in a ballerina and the other during a 9year period in an avid jogger. However, to our knowledge, no reports of more rapid growth of a herniation pit have been presented since that time. We present a case in which the linear dimensions of a pathologically

Report

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