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Introduction: Actinomycosis is a rare and chronic disease
caused by Actinomyces, gram-positive anaerobic bacteria that normally colonize
the human mouth and the digestive and genital tracts. This infection is
extremely uncommon in the oral mucosa membranes and the predisposing factors
may be local and systemic, such immunosuppression.
Case presentation: This paper aims to report the
case of a liver-transplanted patient, with ulcerated and painful lesions on the
back of the tongue, buccal mucosa and lower vestibule fundus, associated to
poor oral hygiene. For diagnostic purposes, the microbiological culture (oral
swab) of the lesions was performed and also an incisional biopsy of the tongue
lesion, confirming the clinical suspicion of Actinomyces infection. Patient was
treated with specific antimicrobial coverage and solutions for mouthwash and,
as an adjuvant, antimicrobial photodynamic therapy (aPDT) was used by the
hospital dental service.
Results: The results of the oral care application and
the PDT show an improvement in the symptoms and good clinical recovery of the
oral tissue.
Conclusion: Photodynamic antimicrobial therapy (aPDT) is
an adjunctive therapeutic modality in the treatment of oral bacterial
infectious manifestations, with satisfactory results.
Keywords: Wound infection, Actinomycosis, Lasers,
Photoch
INTRODUCTION
Actinomycosis
is a rare disease caused by a microorganism called Actinomyces, gram-positive
anaerobic bacteria that normally colonizes the human mouth and the digestive
and genital tracts. Although Actinomyces is naturally present in the oral
microbiota, in some cases it may become pathogenic [1].
This
infection is anatomically and clinically divided into three types:
cervicofacial, pulmonary and abdominal, the first being the most common form, but,
in the oral cavity, this infection is considered rare [1,2].
When in the oral cavity, this infection can affect
both soft and bone tissue and its appearance is usually associated with trauma
or previous infections. In most cases, patients exhibit classic symptoms of
abscess and formation of sulfur granules is indicative of actinomycosis [3].
Clinical diagnosis can sometimes be
problematic as this pathology may present clinical features similar to fungal
infections or malignant tumors. Among the diagnostic methods, culture is the
gold standard in the diagnosis of this condition [1,3].
Patients with actinomycosis require a
prolonged antibiotic scheme (6 to 12 months) and in high doses, being
Penicillin G or Amoxicillin considered the drugs of choice for the treatment of
this lesion. Preventive measures, such as the reduction of alcohol consumption and
the improvement of
Photodynamic antimicrobial therapy (aPDT) is
a promising co-adjuvant treatment method for the eradication of microorganisms
in the oral cavity [5,6]. The mechanism of photosensitization of aPDT consists
of the interaction of light with the photosensitizer and oxygen, generating
free radicals that induce severe damage to the microbial cells, leading to its
death [7].
The aPDT is not considered a substitute for
antimicrobial drugs or any conventional treatment, but rather an important
modality of complementary treatment of localized oral infections, especially in
cases of resistant microorganisms. The sensitivity of bacteria, viruses and
fungi to PDT suggests its applicability in localized shallow infections and
known microbiota. Low cost, minimal side effects and reduction of the
probability of recurrence are the main advantages of the therapy, besides
technical simplicity and absence of risk of microbial resistance [6-9].
Health professionals should be aware of the
possibility of actinomycosis, always aiming at a definitive and timely
diagnosis and the implementation of an effective treatment protocol [2]. In
view of this, this paper proposes to report a rare case of a patient diagnosed
with actinomycosis in the oral cavity.
CASE PRESENTATION
This is a descriptive study, framed as a case
report. The patient agreed to the disclosure of data and images by signing the
Informed Consent Form, being aware that information is unique and exclusively
for scientific purposes, while preserving the patient’s anonymity in full.
Patient NJE, 69 years old, male, ex-smoker
(stopped 30 years ago), ex-alcoholic (stopped 30 years ago), clinical picture
of ulcerative colitis since 2000 and primary sclerosing cholangitis, submitted
to liver transplantation in the year 2006 and using immunosuppressive
medication since then (Tracolimus 2 mg/day). With complaint of bloody diarrhea
for 4 months associated with abdominal pain and recurrent abdominal distension.
He was treated with hydrocortisone, presenting improvement of the clinical
picture, but with recurrence after discontinuation of the medicine. The patient
was then admitted to the Oswaldo Cruz University Hospital of the University of
Pernambuco (HUOC/UPE) for research and treatment/control of the clinical
picture.
During the hospital stay, NJE complained of
pain and wounds in the oral cavity, being requested the opinion of the
dentistry team of CEON/HUOC/UPE. The presence of ulcerated lesions on the back
of the tongue (Figure 1A), right
buccal mucosa (Figure 1B) and lower
right vestibule fundus (Figure 1C),
as well as unsatisfactory oral hygiene, were observed. In view of the clinical
findings and medical history, the diagnostic hypothesis was suggestive of oral
infectious manifestation. Microbiological analysis (oral swab) of oral lesions
was indicated and carried out, as well as an incisional biopsy on the tongue
lesion. The clinical-surgical procedure occurred under local anesthesia,
without intercurrences. After the procedure, the patient was advised about the
importance of oral hygiene and it was prescribed the use of toothbrushes with
small heads, soft bristles and non-abrasive toothpaste, and also mouthwash with
sodium bicarbonate solution (8/8 h), chlorhexidine digluconate 0.12% without
alcohol (12/12 h) and local application of hydrogen peroxide 10 volumes, with
the aid of a gauze, on the lesions (12/12 h).
The dental team of CEON/HUOC/UPE followed NJE daily. The patient
reported, after 72 h of the institution of oral care protocol, improvement of
painful symptoms in oral cavity. After 14 days of the procedure for oral
diagnosis, microbiological culture of oral lesions and incisional biopsy of the
tongue were conclusive for the diagnosis of infection by Actinomyces spp. (Report No. 172445), this microorganism was also
found in biopsy and culture of ulcerated lesion in rectum (Report No.
260485-SC). With the conclusive diagnosis, specific antimicrobial coverage was
established for the systemic infectious disease and, as an adjuvant in the
treatment of oral lesions, photodynamic antimicrobial therapy (aPDT) was
indicated. After signing the Term of Consent and Authorization, the aPDT was
performed, with a 24 h interval between sessions. The aPDT protocol consisted
of the initial application of the photosensitizer (methylene blue - 0.05%) on
all oral lesions and, after 5 min of interaction of the dye with the tissue,
the low-level laser (GaA1As and InGaAlP) was applied (MMOptics device, Brazil),
following the manufacturer’s specifications: the visible red spectrum (660 nm),
with fixed power (output) of 100 mW, spot size de 0.03 cm2 and
energy density of 6 J/cm2 and exposure time per point: 60 s. The
mode of application was of the punctual type, until it contemplated all the
area of the lesions (Figures 2A-2C).
With continuous use of the mouthwash with chlorhexidine digluconate 0.12%
(31 days), a blackened pigmentation appeared in oral mucosa, mainly on the back
of the tongue being then suspended the use of this
mouthwash solution. After suspension, the oral mucosa returned to its normal
appearance (Figure 3).
The oral clinical picture was then controlled and treated daily with
satisfactory resolution of the pains symptoms in addition to good clinical
tissue recovery (Figures 4A-4C), but despite this, due to other systemic
infectious complications, such as Trichosporon Asahii infection, the patient
died.
DISCUSSION
Actinomycosis is an extremely rare infectious disease in the membranes of
the oral mucosa and its clinical presentation is usually characterized by
suppurative granulomatous inflammation, with abscess and fistula [2,4,10]. The
reported case demonstrates unusual features of the oral infectious
manifestation by Actinomyces, with ulcerative lesions on the back of the
tongue, buccal mucosa and lower vestibule fundus. However, other cases found in
the literature also present a clinical manifestation that may evade the pattern
[2].
Predisposing factors for the onset of this condition may be local or
systemic, such as poor oral hygiene, trauma, surgical procedures, diabetes,
long-term corticosteroid therapy and immunosuppression. The risk of developing
the disease is also increased in smokers and alcoholics [1,3]. In the case
reported, the patient presented a history of cigarette and alcohol consumption
and also underwent medical treatment based on systemic corticosteroids.
Another predisposing factor presented by the patient is related to
immunosuppression. Transplanted patients need to use several immunosuppressive
agents throughout their post-transplant life and may present an increased risk
for the development of opportunistic infections, whether of viral or bacterial
origin [11]. The patient underwent liver transplantation in the year 2006 and
since then makes daily use of Tracolimus, which is an immunosuppressive
medication, usually used with the aim of reducing the risk of rejection of the
transplanted organ. Then, faced with this immunosuppression, the patient became
more susceptible to the development of infectious processes, such as
actinomycosis.
Treatment with systemic antimicrobials is the therapy of choice for
bacterial infections by Actinomyces [3,12]. However, in the literature, the
number of studies that show antimicrobial photodynamic therapy as effective in
the adjuvant treatment of infectious conditions has been increasing. The aPDT
presents as promising, with several applications and numerous advantages, among
them, the absence of side effects and the impossibility of resistance acquired
by bacteria [6,7,13].
The use of methylene blue dye has been Communty-Europen (CE)-licensed for
use in dental medicine and maxillofacial surgery in Europe since 2003 and has
been used for the treatment of infected wounds. The authors have used the
technique in clinical practice for medication-related osteonecrosis of the jaw
lesions for at least 10 years for more than 200 patients in combination with
systemic antimicrobial therapy with b-lactam antibiotics combined with
b-lactamase inhibitors. They have found marked improvement especially in wounds
with microbiologically proven colonization with Actinomyces species [13].
The first group to report the use of mouse wound infection models to
investigate the effects of PDT in treating excisional wound infected with Escherichia coli and Pseudomonas aeruginosa were Hamblin et
al. [14]. The experiment consisted in a single wound realized on the backs of
healthy mice and infected with a suspension of bioluminescent bacteria
transduced with a plasmid containing a lux gene operon, which permitted to
monitor the infection in real time by a sensitive charge-coupled camera. In
PDT-treated mice, the authors observed a light dose dependent loss of
luminescence with a 99% reduction after the four light aliquots, which were not
seen in the untreated wound.
Regarding the efficiency of bacterial inactivation by aPDT, it is known
that Gram-positive bacteria are generally more susceptible to aPDT than
Gram-negative species. These differences can be explained by structural
differences in cell walls [15]. As the infectious disease in question is caused
by Gram-positive bacteria, it is even more appropriate to perform the aPDT as a
therapeutic modality associated with systemic antimicrobials.
The exact nature of the microbial killing caused by different types of
lasers emitting light in the intermediate wavelength region has not yet been
determined. The laser beam effect on bacterial cells depends on laser
parameters (wavelength, power, time and mode of emission, beam profile and spot
size) and the characteristics of each bacterium [16].
In the scientific databases, in vitro clinical studies and case reports
demonstrating the use of the laser through aPDT in the microbial inactivation
of several gram positive and negative bacteria can be found, for example in
cases of Pseudomonas aeruginosa, Fusarium sp. and others [13,14,16].
However, the use of this therapy in actinomycosis disease with oral infections,
being then a promising therapy, used in a rare oral manifestation, with few
cases described in the literature.
Therefore, health professionals should be aware of the existence of
infectious diseases in the mouth, which may even mimic malignant conditions. It
is extremely important that the diagnosis is correctly established so that the
treatment adopted is consistent with the condition exhibited by the patient
[1].
CONCLUSION
Infections are the leading causes of morbidity and mortality in liver
transplant patients. The clinical diagnosis associated with microbiological
culture is fundamental for confirmation of the actinomycosis disease.
Infectious oral lesions by actinomyces are rare and specific antibiotic therapy
is the treatment of choice for these cases. Photodynamic antimicrobial therapy
(aPDT) is an adjunctive therapeutic modality in the treatment of oral bacterial
infectious manifestations, with satisfactory results.
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