BritishJournalofOphthalmology 1993; 77:824-826

A rare case of orbital mucormycosis with gas

gangrene panophthalmitis

Vinod K Gupta, Waleed Al-Tuwarqui

Mucormycosis is a disease caused by a fungusfrom the Phycomycetes class and the Mucoralesorder. Phycomycetes are ubiquitous fungi occur-

ring in soil, air, skin, body orifices, manure, andfood. They are commonly seen in diabetics,particularly in patients with ketoacidosis.'"Numerous other predisposing conditions havebeen reported.2 36Gas gangrene refers to a necrotising infection

of the soft tissue caused by a Clostridium species.Clostridium perfringens is recovered in about 80%of cases. Other Clostridium species which com-

monly cause gas gangrene in humans are nozyi,septicum, and sordellii.7

Case reportA 76-year-old Saudi Arabian man was admittedto the coronary care unit of the Security ForcesHospital with acute myocardial infarction. The-patient had been taking oral hypoglycaemic

Figure I Left orbital cellulitis with marked proptosis.

OphthalmologyDepartment, SecurityForces Hospital, PO Box3643, Riyadh 11481,Saudi ArabiaV K GuptaW Al-TuwarquiCorrespondence to:Dr V Gupta.Accepted for publication28 July 1993

Figure 2 Computed tomography scan showing left orbit fullofgas with loss ofdefinition oforbital contents and left atrophiceyeball pushedforward.

drugs for diabetes mellitus for the past 12 years;on admission this was changed to insulin. He wasalso on oral steroids for his bronchial asthma.The left eye had been totally blind for the past 10years and he had previously had glaucomasurgery. His right eye was pseudophakic.On the fifth day of admission the patient

suddenly developed painless left proptosis andlid swelling. There was no history of trauma butmild epistaxis on the third day of admission hadbeen noted. On initial ocular examination, therewas severe panophthalmitis, orbital cellulitis,and proptosis with lid oedema (Fig 1). Foulsmelling brownish pus with gas bubbles wasexuding from the cornea and conjunctiva.Ocular motility was absent. An urgent computedtomography (CT) scan of the orbit and brainshowed the orbit to be full of gas, with loss ofdefinition oforbital contents, and the left eye ballwas pushed forward (Fig 2).

Within hours, the patient also became febrilewith marked periorbital pain. Gas gangrene ofthe orbit was considered. A Gram stain of orbitalcontents aspirated with an 18 gauge needleshowed numerous long Gram positive bacilli. Nofungi were seen. On the same day, under generalanaesthesia, a left orbital exenteration was per-formed. Gas bubbles were coming out of thenecrosed orbital tissues accompanied by a veryfoul smell and the surgery was almost bloodless.The orbital walls were intact and seemedhealthy. The orbital contents were cultured andcytological and histological examination carriedout.The antibiotics were changed to pipracillin 2 g

intravenously at 6 hourly intervals and penicillinG 3 mega units intravenously, also every 6 hours.The patient became afebrile and generallylooked better.

Histological examination showed numerousbroad non-septate, irregularly branching, thickwalled hyphae in the necrotic tissue (Fig 3), theoptic nerve sheath, and also within the vascularlamina. The hyphae were consistent withzygomycetes (Phycomycetes mucor species). AGram stain of the tissue revealed numerousGram positive bacilli (Fig 4), both in the orbitand ocular tissue. However, the fungi were onlyseen in the orbit. The culture on neomycin sheepblood agar plates, incubated in an anaerobicchamber, revealed heavy growth of Clostridiumsordellii. Further identification of Clostridiumspecies was done by using an Anident system kit.No other bacteria had grown within the culture.Urine and blood cultures were negative forClostridium and other bacteria. Clinically, gasgangrene or mucormycosis was not seen in otherparts of the body.Soon after the histology revealed mucormyco-

sis the patient was put on amphotericen B 60 mg

824 on M

arch 7, 2020 by guest. Protected by copyright.

http://bjo.bmj.com

/B

r J Ophthalm

ol: first published as 10.1136/bjo.77.12.824 on 1 Decem

ber 1993. Dow

nloaded from

A rare case oforbital mucormycosis with gas gangrene panophihalmitis

4:A

Figure 3 Mucormycosis inorbital tissue. Broad thickwalled, branching wn- intravenously at 6 hourly

iseptate hyphae cultured on dextrose agar p

(haematoxylin and eosin for fungi. Forty eight hoursstain x 140). patient developed a fever, co

sided hemiparesis. A CT sshowed infarction of the lefinternal capsule. Examinatishowed an extension of the in

palate. Unfortunately, the Xdeteriorated and he developec6 days after the operation. T.on mechanical ventilation.cardiac arrest on the ninth po;

did not respond to cardiopu]tion and died.

CommentMucormycosis is notable forand mortality rates. The dise:fests itself as unilateral faciainternal and external ophthahoptosis, and severe visual loss.The nose and the paranasal

the initial sites of infection. Ocan occur by a contiguous slnormal foramina in the media

Figure 4 Numerous large ing the blood vessels. Most p;Gram positive bacilli, orbital mucormycosis haveClostridium, seen in ocularand orbital tissue (Gramstaining).

8 ;S - 4 ..................... ; _

APE

-.

W ~~

systemic disease. Mucormycosis has beenreported occasionally in otherwise healthy indi-viduals." Multiple risk factors were present inthis case including diabetes mellitus, systemiccorticosteroids, and perhaps recent acute myo-cardial infarction.Management of orbital mucormycosis

depends on early diagnosis, control of anyunderlying disease, systemic antifungal therapy,and aggressive surgical debridement."'368-1

ve*.jf t'' .t Amphotericin B was first introduced into clinicaluse in phycomycosis in 1955 by Harris" andstill seems to be the drug of choice.

Aggressive surgical debridement includingexenteration is necessary in most cases. How-ever, Bullock et al3 reported two cases of rhino-

4 orbital mucormycosis successfully treated with-out exenteration with the sparing of vision in onecase. Kohn and Hepler6 reported eight cases ofrhino-orbital mucormycosis managed success-

ntervals. Contents fully without exenteration. In 1989, Luo et al'°Lates were negative reported a case of orbital mucormycosis withafter surgery, the retinal and ciliary artery occlusion. It was treatednfusion, and right successfully without exenteration.scan of the brain Gas gangrene panophthalmitis usually follows't frontal lobe and after penetrating trauma, with or without aion of the throat retained intraocular foreign body.'3-" The classicifection to the hard cause of this infection was nicely summarised by?atient's condition Duke-Elder."4 Leavelle"3 in 1955 reviewed 53d respiratory arrest previously reported cases and also added threehe patient was put cases of his own. In 1974, Frantz et al'8 reportedHe had a second the only known case of Clostridium perfringensstoperative day but endophthalmitis produced by endogenousImonary resuscita- spread. So far, almost all the cases were treated

by enucleation or evisceration. Crock et al,'9 in1985, reviewed the literature up to 1983 andadded a case of their own. They described apatient with a retained globe and useful visual

its high morbidity acuity of 20/60 after an ocular infection withase typically mani- Clostridium perfringens. This patient had bilateralil pain, headache, ocular trauma. The right eye was enucleated andmoplegia, blephar- an aqueous sample from the left eye showed.1 3-6 Clostridium perfringens. Wiles and Ide'0 reportedsinuses are usually a case of Clostridium perfringens endophthalmitistrbital involvement following penetrating trauma: lensectomy andpread through the subsequently vitrectomy improved vision tod wall or by invad- 20/40. Kelly and Steahly" reported a case ofatients with rhino- successful prophylaxis of Clostidium perfringens

a predisposing endophthalmitis. This patient had received apenetrating eye injury with a nail contaminatedwith manure. After lensectomy and vitrectomythe patient was given an intravitreal antibioticinjection and his final corrected vision was 20/25.These are the only three cases of treatedClostridium perfringens endophthalmitis withretained useful vision.

It seems that our patient may be the onlyknown case of Clostridium sordellii panophthal-mitis with orbital cellulitis. In the spore form,

- Clostridium is resistant to many disinfectantagents. We believe this patient had intraocularClostridium spores from previous surgery or,possibly, an old injury. Mucormycosis is knownto invade the walls of the blood vessels and tocause occlusion, thrombosis, and infarction.Clostridium is known to thrive in tissues with lowoxygen tension. In this case, recent mucormyco-

-^ sis vascular occlusion caused a tissue hypoxiawhich helped in fulminating Clostridium growthand spread.

825 on M

arch 7, 2020 by guest. Protected by copyright.

http://bjo.bmj.com

/B

r J Ophthalm

ol: first published as 10.1136/bjo.77.12.824 on 1 Decem

ber 1993. Dow

nloaded from

Gupta, Al-Tuwarqui

1 Ferry AP, Abedi S. Diagnosis and management of rhino-orbitocerebral mucormycosis: a report of 16 personallyobserved cases. Ophthalmology 1983; 90: 1096-104.

2 Edward JE. Zygomycosis. In: Hoeprich PD, ed. Infectiousdiseases. 3rd ed. Philadelphia: Harper and Row, 1983:1125-32.

3 Bullock JD, Jampol LM, Fezza AJ. Two cases of orbitalphycomycosis with recovery. Am Ophthalmol 1974; 78:811-5.

4 Gass JDM. Acute orbital mucormycosis: report of two cases.Arch Ophihalmol 1%1; 65: 214-20.

5 Schwartz JN, Donnelly EH, Klintworth GK. Ocular andorbital phycomycosis. Surv Ophthalmol 1977; 22: 3-28.

6 Kohn R, Hepler R. Management of limited rhino-orbitalmucormycosis without exenteration. Ophthalmology 1985;92: 1440-4.

7 Wolfe BM. Gas gangrene. In: Hoeprich PD, ed. Infectiousdiseases. 3rd ed. Philadelphia: Harper and Row 1983:1343-7.

8 Baum JL. Rhino-orbital mucormycosis occurring in an other-wise healthy individual. AmJ Ophthalmol 1967; 63: 335-9.

9 Zak SM, Katz B. Successfully treated spheno-orbital mucor-

mycosis in an otherwise healthy adult. Ann Ophthalmol 1985;17:344-8.

10 Luo QL, Orcutt JC, Seifter LS. Orbital muconnycosis withretinal and ciliary arterv occlusion. BrJ Ophthalmol 1989;73: 680-3.

11 Blitzer A, Lawson W, Meyers BR. Patient survival factor inparanasal sinus mucormycosis. Laryngoscope 1980; 90: 635-48.

12 Harris JS. Mucormycosis. Paediatrics 1955; 16: 857.13 Leavelle RB. Gas gangrene panophthalmitis. Arch Ophthalmol

1955; 53:634.14 Duke-Elder S. System of ophthalmology. Vol 14. London:

Kimpton, 1972: 405-10.15 Levitt JM, Stam J. Clostridium perfringens panophthalmitis.

Arch Ophthalmol 1970; 84: 227-8.16 Bhargava SK, Chopder A. Gas gangrene panophthalmitis.

BrJ Ophthalmol 1971; 55: 136-8.17 Wilk CM, Ruprecht KW, Lang BK. Fulminant gas gangrene

panophthalmia following perforating scleral injuries. KlinMonatsbl Augenheilkd 1989; 195: 243-7.

18 Frantz JF, Lemp ML, Font RL, Stone R, Eisner E. Acuteendogenous panophthalmitis caused by Clostridiumperfringens. AmJ Ophthalmol 1974; 78: 295-303.

19 Crock GW, Heriot WJ, Janakireman P, Weiner JM. Gasgangrene infections of eyes and orbits. Br Ophthalmol1985; 69: 143-8.

20 Wiles SW, Ide CH. Clostridium perfringens endophthalmitis[Letter]. AmJ7Ophthalmol 1991; 654-6.

21 Kelly LD, Steahly LP. Successful prophylaxis of Clostridiumperfrigens endophthalnitis. Arch Ophthalmol 1991; 109:1199.

826 on M

arch 7, 2020 by guest. Protected by copyright.

http://bjo.bmj.com

/B

r J Ophthalm

ol: first published as 10.1136/bjo.77.12.824 on 1 Decem

ber 1993. Dow

nloaded from