Oral and maxillofacial myiasis: a case series and literature

review
Antonio Azoubel Antunes, DDS,
a
Thiago de Santana Santos, DDS, MSc,
a
Rafael Linard Avelar, DDS, MSc,
b
Evandro Carneiro Martins Neto, DDS,
c
Bruno Macedo Neres, DDS,
d
and Jos Rodrigues Laureano Filho, DDS, MSc, PhD,
e
So Paulo,
Porto Alegre, and Recife, Brazil
UNIVERSITY OF SO PAULO, PONTIFIC CATHOLIC UNIVERSITY OF RIO GRANDE
DO SUL, OSWALDO CRUZ HOSPITAL, RESTAURAO HOSPITAL, AND
UNIVERSITY OF PERNAMBUCO
Objective. The aim of this study was to describe a series of 10 cases of oral-maxillofacial myiasis, discussing its main
features, demographic distribution, and treatment aspects.
Study design. A retrospective study was carried out involving male and female patients of any age with oral-
maxillofacial myiasis. The sample was determined by spontaneous demand at the emergency ward of a hospital
between January 2005 and January 2011 (6 years). After treatment of each case, data were gathered on the presence
of associated systemic disorders, time elapsed since onset of the disease, and treatment established. A review of the
literature on this topic was also carried out.
Results. The sample was made up of 10 patients, all treated with surgical debridement whether or not associated with
the use of oral ivermectin. Mean time elapsed since the onset of the disease ranged from 4 to 36 months. The middle
third of the face was the most frequently affected region (7 cases). Oral-maxillofacial myiasis predominantly affected
the male gender (70%).
Conclusions. Oral-maxillofacial myiasis affects individuals with poor hygiene and neurologic and/or psychologic
alterations. It occurs predominantly in countries near the tropics. The treatment of choice is surgical debridement.
(Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:e81-e85)
Myiasis is the invasion of human or animal tissue by y
larvae that evolve into parasites.
1
This is a worldwide
phenomenon related to latitude and the life cycle of
various species of ies. Myiasis occurs predominantly
in the tropics and subtropics in countries with inade-
quate hygiene, poor housing conditions (proximity to
domesticated animals), and warm weather.
2
Myiasis is classied as either primary or secondary.
Primary myiasis is caused by larvae that feed on living
tissue (biophagous). This form of myiasis is common in
cattle and rare in humans. It is produced by Cochlio-
myia hominivorax (varejeira y), which lays 20-400
eggs on exposed wounds, with larvae hatching within
24 hours.
3
Secondary myiasis is caused by ies that
feed on dead tissue (necrobiophagous). This is the more
common type and attacks patients with lesions that
have necrotic cavities.
1,4
Infestation is most often subcutaneous, producing
furunculoid or boil-like lesions, and it can also occur in
certain body cavities and wounds.
5
The larvae obtain
their nutrition from the surrounding tissue and burrow
deep tunnels into soft tissue, separating the gums and
mucoperiosteum from the bone.
6
Oral-maxillofacial myiasis has been described in the
literature since 1909 and is considered to be a rare occur-
rence.
7
The cases described are mostly due to medical and
anatomic conditions, such as neglected mandibular frac-
ture,
8
lip incompetence, cerebral palsy, mouth breathing,
anterior open bite,
9
patients undergoing mechanical ven-
tilation,
10
cancrum oris,
8
and tooth extraction.
11
The aim of the present study was to describe a series
of 10 cases of oral-maxillofacial myiasis, discussing its
main features, demographic distribution, and treatment
aspects and comparing the ndings with earlier studies
in the literature.
a
Oral and Maxillofacial Surgeon, PhD Student, University of So Paulo.
b
Oral and Maxillofacial Surgeon, PhD Student, Pontic Catholic
University of Rio Grande do Sul.
c
Senior Resident in Oral and Maxillofacial Surgery, Oswaldo Cruz
Hospital.
d
Senior Resident in Oral and Maxillofacial Surgery, Restaurao
Hospital.
e
Adjunct Profesor, Department of Oral Medicine, University of Per-
nambuco.
Received for publication May 8, 2011; accepted for publication May
28, 2011.
1079-2104/$ - see front matter
2011 Mosby, Inc. All rights reserved.
doi:10.1016/j.tripleo.2011.05.026
e81
MATERIALS AND METHODS
A retrospective study was carried out involving male
and female patients of any age with oral-maxillofacial
myiasis. The sample was determined by spontaneous
demand at the emergency ward of a hospital between
January 2005 and January 2011 (6 years). After treat-
ment of each case, data were gathered on gender, age,
site, species of larva, the presence of associated sys-
temic disorder, time elapsed since onset of the disease,
and treatment established. In all cases, the larvae were
sent for parasitologic analysis for the conrmation of
the species.
All of the patients underwent operations soon after
admission to the emergency ward. Under general anes-
thesia, the mechanical removal of the larvae was per-
formed, along with debridement of the necrotic tissue
and an attempt at the maximal possible primary closure
of the wound, as exemplied in case 1 (Figs. 1-4). The
patients were also treated with a systemic antibiotic
(cephalotin, 1 g every 6 h), antiinammatory, and an-
algesic. No specic medication protocol for ivermectin
was instituted, and the decision to use this drug fell to
the health care professional conducting each case.
Literature review
All papers published in the literature between 1963
and 2010 on oral-maxillofacial myiasis in all languages
were surveyed to determine the mapping of cases per
Fig. 1. Clinical aspect of the patient.
Fig. 2. Necrotic tissue after larvae removal.
Fig. 4. Larvae removed from the wound.
Fig. 3. Primary wound closure after debridment.
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e82 Azoubel Antunes et al. December 2011
country of occurrence of the disease. The Medline
(Pubmed) database was used for this search, using the
following descriptors were used: oral myiasis, nasal
myiasis, ophtalmomyiasis, and maxillofacial myiasis.
Case reports that did not address human myiasis,
those that appeared to be duplicated in the searches
with different descriptors, and those carried out in
countries that no longer exist were excluded. After the
denition of the sample, the cases were counted and
separated by country. Among a total of 570 papers
found in the search, 490 were excluded owing to du-
plication in the searches using different descriptors and
for reporting cases of myiasis in sites other than the
oral-maxillofacial region. Two were excluded for the
fact that they were from countries that no longer exist
(Czechoslovakia) and one was excluded because the
origin of the paper could not be identied. Papers that
reported cases in animals were also excluded. Thus, a
total of 493 papers were excluded and 77 papers (86
cases) fullled the inclusion criteria, as distributed on
the map in Fig. 5.
RESULTS
The sample was made up of 10 patients, all of whom
were treated with surgical debridement whether or not
associated with the use of oral ivermectin. Mean time
elapsed since the onset of the disease ranged from 4 to
36 months. The middle third of the face was the most
frequently affected region (7 cases). Oral-maxillofacial
myiasis predominantly affected the male gender (70%).
These data are presented in Table I.
DISCUSSION
Oral-maxillofacial myiasis is a rare condition caused
by ies of the order Diptera, which lay eggs or larvae in
food, necrotic tissue, wounds, and intact mucosa. Clas-
sication is based on location in the host body [dermal,
subdermal, nasopharyngeal, internal (organs), or uro-
genital]; in parastologic terms, the condition is classi-
ed based on the parasite/host relationship (obligatory
or facultative).
12
The parasitologic examination of larval samples re-
moved from the present patients determined that the
etiologic agent in all cases was Cochliomyia hominiv-
orax of the family Callipharidae, which is found mainly
in Central and South America.
13
Its larvae prefer warm-
blooded animals or humans as hosts. The ies feed on
exudates and deposit eggs in necrotic tissue. Each fe-
male is capable of laying 2,800 eggs, which are pref-
erentially deposited in a recent wound, lesion, or simple
scratch.
14
In 12-24 days after being laid, the eggs hatch
and the larvae begin to burrow into living tissue, in-
cluding cartilage and bone.
12
The complete cycle takes
24 days.
14
A number of authors state that myiasis occurs with
greater frequency in regions with a warm climate in
individuals with inadequate hygiene and poor hous-
ing conditions (proximity to domesticated ani-
mals).
2,9,15
The mapping of the cases reported in the
literature revealed a predominance of cases in trop-
ical and subtropical regions. The countries with the
largest number of cases were Brazil (21 cases), India
(16 cases), and Turkey (11 cases). Among the 86
cases of oral-maxillofacial myiasis reported in the
international literature, 21 (24.4%) occurred in Bra-
zil, which is the country of origin of the present
study. All patients exhibited inadequate hygiene and
associated comorbidities.
A greater frequency of the disease is reported in
patients with an altered psychologic prole or neuro-
logic disease associated with predisposing clinical con-
ditions, such as lip incompetence, mouth breathing,
anterior open bite, under mechanical ventilation, or a
wound that did not receive adequate care.
8-11
In the
cases reported, 6 patients has some type of psychologic
or neurologic disorder, which, in theory, made them
more susceptible to negligence regarding general health
and progression of the disease.
The treatment of choice for oral-maxillofacial my-
iasis is surgical debridement, which may or may not
be associated with the use of a systemic medication.
Debridement has a curative function, but some for-
eign-body reactions may occur in cases in which
larvae remain in the surgical wound owing to incom-
plete debridement.
5
Certain drugs are suggested as
adjuvants to treatment, such as ivermectin and nitro-
furazone.
16
Ivermectin is a semisynthetic agent from the family
of macrolides, synthesized from natural substances,
such as avermectin (obtained from actinomycetes). In-
vermectin is a broad-spectrum antiparasitic drug for
veterinary use, but with proven efcacy for some par-
Fig. 5. World distribution of the 86 cases of oral and maxil-
lofacial myiasis reported in the literature.
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Volume 112, Number 6 Azoubel Antunes et al. e83
asites that attack the human organism. It is generally
administered in a single dose of 150-200 g/kg of body
weight. Invermectin is absorbed quickly and reaches a
high concentration in the blood within a relatively short
period of time.
14
The most common method for dealing with cutane-
ous myiasis is occlusion of the central ulcer to asphyx-
iate the larvae. Spontaneous expulsion of the larvae
occurs soon after the administration of the asphyxiating
agent, which brings larvae deposited in deeper tissue
layers to the surface, thereby facilitating their surgical
removal.
17
Nitrofurazone is a synthetic nitrofuran classied as
a topical broad-spectrum antiinfective agent used
primarily in burn patients, skin grafts, and the pre-
vention of urinary tract infection due to catheter use.
Its specic administration for myiasis involves the
liberal placement of the agent in the wound to allow
the submerged larvae to come into contact with the
offensive liquid substance, forcing them to exit the
interior of the tissue and facilitating their removal.
The topical administration of this agent occurs over
3 consecutive days.
14
In the present study, 5 of the 10 cases were treated
with oral ivermectin as an adjuvant to surgical de-
bridement. Removal of the larvae was successful in
all cases and no clinical difference was observed
between the cases with and without the use of the
drug. A number of authors recommend the use of
ivermectin without surgical debridement.
3,14
How-
ever, this treatment plan was not adopted in any of
the cases reported here.
Based on the ndings of the present study, oral-
maxillofacial myiasis is a disease that affects individ-
uals with poor hygiene, many of whom have neurologic
and/or psychologic alterations. This condition occurs
predominantly in countries near the tropics. The treat-
ment of choice is surgical debridement for the removal
of the cause, which may or may not be associated with
a specic systemic medication.
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Table I. Patient distribution
Patient Gender-age (y) Site Associated disorder Causative agent Duration Treatment
1 M-72 Upper lip Uncontrolled diabetes Cochliomyia hominivorax 36 mo Debridement
2 F-82 Hard palate AIDS mentally ill Cochliomyia hominivorax 6 mo Debridement
ivermectin
3 M-39 Preauricular SAH depression Cochliomyia hominivorax 24 mo Debridement
ivermectin
4 M-67 Retroauricular Diabetes mouth breather Cochliomyia hominivorax 12 mo Debridement
5 F-59 Maxilla Squamous cell carcinoma Cochliomyia hominivorax 24 mo Debridement
6 F-78 Retroauricular Uncontrolled diabetes Cochliomyia hominivorax 8 mo Debridement
ivermectin
7 M-26 Upper lip Mental retardation Cochliomyia hominivorax 3 mo Debridement
8 M-34 Maxilla Mentally ill Cochliomyia hominivorax 18 mo Debridement
Invermectin
9 M-37 Right orbita Retinoblastome depression Cochliomyia hominivorax 6 mo Debridement
Invermectin
10 M-40 Retroauricular Depression Cochliomyia hominivorax 4 mo Debridement
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e84 Azoubel Antunes et al. December 2011
14. Gealh WC, Ferreira GM, Farah GJ, Teodoro U, Camarini ET.
Treatment of oral myiasis caused by Cochliomyia hominivorax:
two cases treated with ivermectin. Br J Oral Maxillofac Surg
2009;47:23-6.
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myiasis. J Infect Public Health 2010;3:43-5.
Reprint requests:
Antonio Azoubel Antunes
Avenida Caramuru, 732, Apto 102A
Repblica Ribeiro Preto SP
CEP: 14030-000
Brazil
antunesctbmf@yahoo.com.br
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