use of ivermectin in the treatment of orbital myiasis caused by cochliomyia hominivorax

3
Lactobacillus, such as L. casei and L. rhamnosus, are the most commonly associated with IE (6, 7). The only report in the English literature of an infection caused by Lactobacillus gasseri describes a case of pyelone- phritis and bacteremia secondary to ureteral obstruc- tion (8). Lactobacillus gasseri was first identified by Lauer and Kandler in 1980 (9). It is a member of the family Lactobacillaceae. It is a Gram positive, non-spore- forming rod, acid tolerant, catalase negative, oxidase negative, non-H 2 S-producing organism, resistant to vancomycin, with a DNA base composition of less than 53% G /C. Lactobacillus gasseri is homofer- mentative, i.e. it forms lactic acid from glucose as the major fermentation product. It grows best under anaerobic conditions, and it can be enumerated on a variety of selective culture media. LAMVAB and Rogosa are the most commonly used selective agars for the isolation of Lactobacillus gasseri. High acetate and low pH of Rogosa agar theoretically permits growth of lactobacilli but suppresses many other strains of lactic acid bacteria. Recent advances in molecular biology including PCR-based methodology have allowed more reliable identification of lactobacilli (10). Lactobacilli species can be discriminated on the basis of known sequence differences in the bacterial 16s rDNA gene (11) using PCR, single-stranded conformational polymorphism (SSCP), and DNA sequencing. CONCLUSION The current case is the first reported case of Fournier’s gangrene caused by Lactobacillus gasseri. Lactobacil- lus gasseri was grown on Rogosa agar and the species level of identification was performed using molecular biologic techniques to sequence the bacterial 16s rDNA gene. We postulate that the patient’s underlying cirrhos and chronic candidal skin infection and local moisture caused breaks in the integument and predis- posed him to this severe infection caused by a commensal organism. REFERENCES 1. Eke N. Fournier’s gangrene: a review of 1726 cases. Br J Surg 2000; 87: 718 /28. 2. Morpurgo E. Fournier’s gangrene. Surg Clin North Am 2002; 82: 1213 /24. 3. Johnin K, Nakatoh M, Kadowaki T, Kushima M, Koizumi S, Okada Y. Fournier’s gangrene caused byCandida species as the primary organism. Urology 2000; 56: 153. 4. Mitsuoka T. The human gastrointestinal tract. In: Wood BJB, ed. The Lactic Acid Bacteria: Volume 1, The Lactic Acid Bacteria in Health and Disease. Essex, England: Elsevier Science Publishers, Ltd; 1992. p. 69 /114. 5. Salminen MK, Rautelin H, Tynkkynen S, Poussa T, Saxelin M, Valtonen V, Jarvinen A. Lactobacillus bacteremia, clinical significance, and patient outcome, with special focus on probiotic L. rhamnosus GG. Clin Infect Dis 2004; 38: 62 /9. 6. Sandre RM, Shafran SD. Infective endocarditis: review of 135 cases over 9 y. Clin Infect Dis 1996; 22: 276 /86. 7. Saxelin M, Chuang N-H, Chassy B, Rautelin H, Ma ¨kela ¨ PH, Salminen S, Gorbach SL. Lactobacilli and bacteremia in southern Finland, 1989 /1992. Clin Infect Dis 1996; 22: 564 /6. 8. Dickgiesser U, Weiss N, Fritsche D. Lactobacillus gasseri as the cause of septic urinary infection. Infection 1984; 12: 14 /6. 9. Lauer E, Kandler O. Lactobacillus gasseri sp. Nov., a new species of the subgenus Thermobacterium.Zbl. Bakt. Hyg. I Abt Orig C 1980; 1: 75 /8. 10. Walter J, Hertel C, Tannock GW, Lis CM, Munro K, Hammes WP. Detection of Lactobacillus, Pediococcus, Leuconostoc and Weissella species in human feces by using group-specific PCR primers and denaturing gradient electrophoresis. Appl Environ Microbiol 2001; 67: 2578 /85. 11. McOrist AL, Jackson M, Bird AR. A comparison of 5 methods for extraction of bacterial DNA from human fecal samples. J Microbiol Methods 2002; 50: 183 /91. Submitted April 22, 2004; accepted May 11, 2004 DOI: 10.1080/00365540410015916 Use of Ivermectin in the Treatment of Orbital Myiasis Caused by Cochliomyia hominivorax PAULO DE TARSO, P. PIERRE-FILHO, NILZA MINGUINI, LEANDRO M. PIERRE and ALESSANDRA M. PIERRE From the Department of Ophthalmology, School of Medical Sciences, State University of Campinas (UNICAMP) , Campinas, Sa ˜o Paulo, Brazil Orbital myiasis cases by dipterous fly larvae are rarely reported. A case of massive orbital myiasis caused by Cochliomyia hominivorax resulting in complete loss of the globe is described. Oral ivermectin was used successfully to eliminate the larvae prior to enucleation, making the mechanical removal of larvae an unnecessary procedure. Scand J Infect Dis 36 Case Reports 503 Scand J Infect Dis Downloaded from informahealthcare.com by Michigan University on 11/12/14 For personal use only.

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Page 1: Use of Ivermectin in the Treatment of Orbital Myiasis Caused by Cochliomyia hominivorax

Lactobacillus, such as L. casei and L. rhamnosus, are

the most commonly associated with IE (6, 7). The only

report in the English literature of an infection caused

by Lactobacillus gasseri describes a case of pyelone-

phritis and bacteremia secondary to ureteral obstruc-

tion (8).

Lactobacillus gasseri was first identified by Lauerand Kandler in 1980 (9). It is a member of the family

Lactobacillaceae. It is a Gram positive, non-spore-

forming rod, acid tolerant, catalase negative, oxidase

negative, non-H2S-producing organism, resistant to

vancomycin, with a DNA base composition of less

than 53% G�/C. Lactobacillus gasseri is homofer-

mentative, i.e. it forms lactic acid from glucose as the

major fermentation product. It grows best underanaerobic conditions, and it can be enumerated on a

variety of selective culture media. LAMVAB and

Rogosa are the most commonly used selective agars

for the isolation of Lactobacillus gasseri. High acetate

and low pH of Rogosa agar theoretically permits

growth of lactobacilli but suppresses many other

strains of lactic acid bacteria.

Recent advances in molecular biology includingPCR-based methodology have allowed more reliable

identification of lactobacilli (10). Lactobacilli species

can be discriminated on the basis of known sequence

differences in the bacterial 16s rDNA gene (11) using

PCR, single-stranded conformational polymorphism

(SSCP), and DNA sequencing.

CONCLUSION

The current case is the first reported case of Fournier’s

gangrene caused by Lactobacillus gasseri. Lactobacil-

lus gasseri was grown on Rogosa agar and the species

level of identification was performed using molecularbiologic techniques to sequence the bacterial 16s

rDNA gene. We postulate that the patient’s underlying

cirrhos and chronic candidal skin infection and local

moisture caused breaks in the integument and predis-

posed him to this severe infection caused by a

commensal organism.

REFERENCES

1. Eke N. Fournier’s gangrene: a review of 1726 cases. Br J Surg

2000; 87: 718�/28.

2. Morpurgo E. Fournier’s gangrene. Surg Clin North Am 2002;

82: 1213�/24.

3. Johnin K, Nakatoh M, Kadowaki T, Kushima M, Koizumi S,

Okada Y. Fournier’s gangrene caused by Candida species as the

primary organism. Urology 2000; 56: 153.

4. Mitsuoka T. The human gastrointestinal tract. In: Wood BJB,

ed. The Lactic Acid Bacteria: Volume 1, The Lactic Acid

Bacteria in Health and Disease. Essex, England: Elsevier Science

Publishers, Ltd; 1992. p. 69�/114.

5. Salminen MK, Rautelin H, Tynkkynen S, Poussa T, Saxelin M,

Valtonen V, Jarvinen A. Lactobacillus bacteremia, clinical

significance, and patient outcome, with special focus on

probiotic L. rhamnosus GG. Clin Infect Dis 2004; 38: 62�/9.

6. Sandre RM, Shafran SD. Infective endocarditis: review of 135

cases over 9 y. Clin Infect Dis 1996; 22: 276�/86.

7. Saxelin M, Chuang N-H, Chassy B, Rautelin H, Makela PH,

Salminen S, Gorbach SL. Lactobacilli and bacteremia in

southern Finland, 1989�/1992. Clin Infect Dis 1996; 22: 564�/6.

8. Dickgiesser U, Weiss N, Fritsche D. Lactobacillus gasseri as the

cause of septic urinary infection. Infection 1984; 12: 14�/6.

9. Lauer E, Kandler O. Lactobacillus gasseri sp. Nov., a new

species of the subgenus Thermobacterium.Zbl. Bakt. Hyg. I Abt

Orig C 1980; 1: 75�/8.

10. Walter J, Hertel C, Tannock GW, Lis CM, Munro K, Hammes

WP. Detection of Lactobacillus, Pediococcus, Leuconostoc and

Weissella species in human feces by using group-specific PCR

primers and denaturing gradient electrophoresis. Appl Environ

Microbiol 2001; 67: 2578�/85.

11. McOrist AL, Jackson M, Bird AR. A comparison of 5 methods

for extraction of bacterial DNA from human fecal samples. J

Microbiol Methods 2002; 50: 183�/91.

Submitted April 22, 2004; accepted May 11, 2004

DOI: 10.1080/00365540410015916

Use of Ivermectin in the Treatment of OrbitalMyiasis Caused by Cochliomyia hominivoraxPAULO DE TARSO, P. PIERRE-FILHO, NILZA MINGUINI, LEANDRO M. PIERRE

and ALESSANDRA M. PIERRE

From the Department of Ophthalmology, School of Medical Sciences, State University of Campinas (UNICAMP) ,

Campinas, Sao Paulo, Brazil

Orbital myiasis cases by dipterous fly larvae are rarely reported. A case of massive orbital myiasis caused by Cochliomyia

hominivorax resulting in complete loss of the globe is described. Oral ivermectin was used successfully to eliminate the larvae

prior to enucleation, making the mechanical removal of larvae an unnecessary procedure.

Scand J Infect Dis 36 Case Reports 503

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Page 2: Use of Ivermectin in the Treatment of Orbital Myiasis Caused by Cochliomyia hominivorax

Paulo de Tarso P. Pierre-Filho, Rua Alexandre Fleming, s/n, Departamento de Oftalmologia , Faculdade de Ciencias Medicas,

Universidade Estadual de Campinas (UNICAMP), Campinas �/ SP, CEP 13081-970, Brazil (Tel., fax. �/011 55 19

37887936, e-mail. [email protected])

INTRODUCTION

Ophthalmomyiasis refers to infestations of the eye and

/or ocular adnexa by larvae of the order Diptera, and

represents less than 5% of the cases of human myiasis.

When larvae remain outside the eye, it is termed

ophthalmomyiasis externa, while penetration of the

eye itself is termed ophthalmomyiasis interna (1).

Although orbital involvement is rarely described, the

invasive parasitic larvae almost invariably cause mas-

sive destruction of orbital tissue in neglected patients,

accompanied by marked inflammatory reactions and

secondary bacterial infections (1, 2). We report a case

of massive orbital myiasis caused by Cochliomyia

hominivorax treated with oral ivermectin, followed by

enucleation. At the time of presentation, the intrao-

cular contents were totally destroyed.

CASE REPORT

An 80-y-old man, with a complaint of 5 d of severe pain and swelling

in his left orbital region, was brought in by his friend to the

Department of Ophthalmology, State University of Campinas. He

had been deserted by family members 4 y earlier and lived by himself

in a rural area. The patient’s medical history was significant for

diabetes and hypertension. He also was known to be alcoholic and a

chronic smoker.

The clinical examination showed no light perception in the left

eye. Marked left periorbital oedema with multiple cutaneous

erosions was noted. The eyeball was perforated, and maggots were

visualized within the ocular cavity (Fig. 1). The maggots were

identified as larvae of Cochliomyia hominivorax. Computed tomo-

graphic scanning of the orbit revealed extensive soft tissue swelling

and destruction of the left globe. The right globe was unremarkable.

Laboratory findings were within normal limits. He was given a single

oral dose of ivermectin (200 mg/kg), and intravenous cefazolin was

started for a possible secondary infection. A complete resolution of

the myiasis was observed in a 48-h period. The orbital swelling

showed continuous improvement. Enucleation was performed in the

left eye. No larvae were observed during the surgery. The patient was

discharged 2 d later.

DISCUSSION

Ophthalmomyiasis is considered a rare, life-threaten-

ing condition, and can cause sequelae ranging from

minor irritation to blindness, disfigurement, and even

death. The medical consequences vary, and depend in

part on whether the maggots are obligatory or

facultative parasites. Facultative species tend to pro-

duce mild infestations, while obligatory species can be

invasive (3).

The most common cause of ophthalmomyiasis is

the sheep botfly Oestrus ovis. Cochliomyia homini-

vorax, the aetiology in our patient, is less common (4).

Orbital myiasis is, fortunately, the least common form

of ophthalmomyiasis (1). By consulting Pub. Med., we

found that only 13 orbital myiasis patients have been

reported in the last 35 y.

Patients with orbital myiasis have been mostly either

children or elderly demented patients who do not

adequately care for themselves (1, 2). Our patient was

of a low socioeconomic status and had poor hygiene

habits, which may have predisposed to larval invasion.

Debilitating conditions including diabetes mellitus,

hypertension, alcoholism and a prolonged state of

unconsciousness in a rural location contributed to the

fast destruction of the orbital contents of the patient.

Cochliomyia hominivorax is an obligatory parasite,

and although considered to be confined to the New

World, reports have identified this parasite in Asia and

Africa. When large numbers of diptera invade deeply

into the orbit, they rapidly destroy the orbital contents

and adjacent tissues, including bone and meninges (3).

Another case of massive orbital myiasis by Cochlio-

myia hominivorax was reported in a Brazilian patient

with basal cell carcinoma of the eyelid (5); orbital

exenteration was performed.

Management of orbital myiasis should be directed

toward removing all invading organisms and control-

ling the almost inevitable secondary infection. Solu-

tions of hydrogen peroxide, chloroform, ether, ethanol,

cocaine and turpentine have been used to kill and

remove the larvae (1, 6, 7). In some cases, exenteration

has been performed to prevent progression of the

infestation (1, 2, 5).

In this patient a single oral dose of ivermectin

(200 mg/kg) led to spontaneous emigration of the

maggots prior to surgery. To our knowledge, this is

the first reported case of orbital myiasis caused by

Cochliomyia hominivorax in which oral ivermectin

was used successfully, making the mechanical removal

of larvae an unnecessary procedure. We suggest that

oral ivermectin should be considered as an alternative

Fig. 1. Appearance of patient with forced eyelid opening showing

invasion of left eye and orbit by maggots.

504 Case Reports Scand J Infect Dis 36

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Page 3: Use of Ivermectin in the Treatment of Orbital Myiasis Caused by Cochliomyia hominivorax

therapy for cases of orbital myiasis. Early diagnosis

and management are important in preventing compli-

cations.

REFERENCES

1. Kersten RC, Shoukrey NM, Tabbara KF. Orbital myiasis.

Ophthalmology 1986; 93: 1228�/32.

2. Caca I, Unlu K, Cakmak SS, Bilek K, Sakalar YB, Unlu G.

Orbital myiasis: case report. Jpn J Ophthalmol 2003; 47: 412�/4.

3. Chodosh J, Clarridge J. Ophthalmomyiasis: a review with special

reference to Cochliomyia hominivorax. Clin Infect Dis 1992; 14:

444�/9.

4. Savino DF, Margo C, McCoy ED, Friedl FE. Dermal myiasis of

the eyelid. Ophthalmology 1986; 93: 1225�/7.

5. Rocha EM, Yvanoff JL, Silva LMS, Prado AP, Caldato C.

Massive orbital myiasis infestation. Arch Ophthalmol 1999; 117:

1436�/7.

6. Baliga MJ, Davis P, Rai P, Rajasekhar V. Orbital myiasis: a case

report. Int J Oral Maxillofac Surg 2001; 30: 83�/4.

7. Balasubramanya R, Pushker N, Bajaj MS, Rani A. Massive

orbital and ocular invasion in ophthalmomyiasis. Can J Ophthal-

mol 2003; 38: 297�/8.

Submitted February 6, 2004; accepted February 17, 2004

DOI: 10.1080/00365540410020136

Cytomegalovirus Appendicitis FollowingAcute Epstein-Barr Virus Infection in anImmunocompetent PatientZA KANAFANI1, AI SHARARA1, NS SHABB2 and SS KANJ1

From the Departments of 1Internal Medicine and 2Pathology and Laboratory Medicine, American University of Beirut, Medical

Center Beirut, Lebanon

Acute appendicitis due to cytomegalovirus is exceedingly rare. It occurs mostly in individuals with the acquired

immunodeficiency syndrome and has been reported once in an immunocompetent patient. This report describes a man who

developed acute appendicitis following acute Epstein-Barr virus infection, and in whom cytomegalovirus was incriminated based

on pathological examination.

S. Kanj, Department of Internal Medicine, Division of Infectious Diseases, American University of Beirut Medical Center, P.O.

Box 113-6044, Hamra 110 32090, Beirut, Lebanon (Tel. �/961 1 350000, fax. �/961 1 370814, e-mail. [email protected])

INTRODUCTION

Infectious mononucleosis (IM) is most commonly

caused by acute Epstein-Barr virus (EBV) infection,

followed by cytomegalovirus (CMV) infection.

Although the illness is typically self-limited, resolving

uneventfully within 1 or 2 weeks, rare complications

may arise. Virtually any organ system can be involved,

leading to diverse clinical manifestations (1�/3). The

following report describes an immunocompetent manwho developed CMV appendicitis in the setting of

acute EBV infection.

CASE REPORT

A 28-y-old previously healthy homosexual man presented to the

emergency unit with high-grade fever and severe sore throat. On

physical examination, his tonsils were erythematous with purulent

exudates and he had a macular rash over the abdomen and torso.

His white cell count was 11,300/mm3 with 41% neutrophils, 53%

lymphocytes, and atypical lymphocytes seen on peripheral blood

smear inspection. Monospot test was positive, and EBV viral capsid

antigen (VCA) IgM by enzyme-linked immunosorbent assay

(ELISA) (ETI-VCA, Diasorin Sp.A., Italy) was �/140. CMV IgM

and IgG by enzyme-linked fluorescence assay (ELFA) (VIDAS,

BioMerieux, France), as well as HIV antibodies were all negative.

One week later, he returned with persistent high-grade fever,

vomiting, and new-onset severe abdominal pain. He had abdominal

distension with direct and rebound tenderness, mostly in the left

lower quadrant. Computed tomography scan of the abdomen

showed periappendiceal abscess formation and free air, suggestive

of bowel perforation. Laparoscopic exploration revealed multiple

collections of purulent material in the pelvis and right lower

quadrant. The appendix was found to be perforated at several sites

with areas of fibrosis. Pathologic examination showed extensive

inflammation at the tip (Fig. 1). Although the inflammatory

infiltrate was predominantly neutrophilic, it was also associated

with lymphocytes, eosinophils and plasma cells. These cells filled the

lumen and extended from the mucosa out to the serosa. Many

cytomegalic cells were present. These were significantly enlarged

endothelial cells with large nuclei containing large eosinophilic

intranuclear inclusions surrounded by a halo. The cytoplasm was

abundant and contained fine granular eosinophilic material (Fig. 2).

These cells were seen mostly at the tip and in the area of the

perforation, and involved all layers of the appendix. Immunohisto-

chemical stains showed these cells to be strongly positive for CMV

and negative for EBV. Bacterial cultures of the intraoperative

specimens were negative. CMV IgG, which was initially negative,

Scand J Infect Dis 36 Case Reports 505

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