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Acta Médica Costarricense
On-line version ISSN 0001-6002Print version ISSN 0001-6012
Acta méd. costarric vol.55 n.1 San José Jan./Mar. 2013
Caso clínico
Rhinoscleroma
John Segura-Vílchez, 1
Paulina González-Rojas, 2 Lissette Retana-Moreira3
Author´s
affiliation:
Abstract
The case
of a 19–year–old male patient
from a rural area is presented. He had a 3-year history of nasal
obstruction, episodes
of facial inflammation, epistaxis, fetid rhinorrhea, hearing loss in the right ear,
bilateral axillary and multiple facial
bilateral lymphadenopathies.
He was referred for consultation to the
A second
biopsy was positive for Klebsiellapneumoniae
subsp. rhinoscleromatis,
described histologically as a pseudoepitheliomatouse
hyperplasia with dense inflammatory infiltrate and the analysis of
lamina propria showed dense inflammatory
infiltrate with
lymphocytes, plasmatic cells, Russell bodies and macrophages with
vacuolated
cytoplasm, with microorganisms and debris. The patient was treated with
oral
ciprofloxacin therapy for seven months, after which the patient was
considered
cured from the etiological point of view.
Keywords:
Klebsiella, chronic granulomatose
disease, biopsy.
been
realized to understand the mechanisms of pathogenicity
of Klebsiella, whose determinants
are mainly
found in capsular antigens.2
K. pneumoniaey K. oxytocaare
the two mainly species of
this genus that cause disease in humans, have also been isolated from
clinical
samples: K. pneumoniae subsp. ozenae, K. pneumoniaesubsp.
rhinoscleromatis,
K. terrigena, K. planticola,
K. ornithinolyticay Enterobacteraerogenes,
which is renamed like K. mobilis.
Masculine
patient, 19 years old, from Guanacaste,
referred to the Otorhinolaryngology
Department (ORL)
of Mexico Hospital, by a severe granulomatous
obstructive lesion of the nasalseptum,
apparent
inflammation of crystalloids bodies, Leishmania
sp and fungi smears were negative; with a biopsy report realized in
February of 2008, which stated as clinical history of a nasal granulomatous lesion reminiscent Leishmania
sp. That report indicates the presence of a chronic inflammatory
infiltrative tissue, predominantly foamy macrophages and many particles
of
crystalloid aspect; without evidence of multinucleated giant cells, or
compatible structures of Leishmania
sp.
The
non-pathologic patient´s
record indicates that works as fencing operator and smokes 5 cigarettes
per day
average, occasional elitism, no medical allergies; the pathologic
record
referred a nasal pain since 3 years ago, facial inflammation episodes, epistaxis, fetid rhinorrhea
and
right low hearing.
In March,
2009 it was detected multiple lymphadenopathies
in the face and in both armpits, as also granulomatous
injury with both nostrils occlusion (to the
lobby of the right ear). It was declared under observation of a lymphoproliferative disease, with a biopsy
programming;
three weeks later, in ORL session, a study established for possible
deep
mycosis versus Klebsiellapneumoniae
subsp.
Rhinoscleromatis infection. Finally, they
took a
biopsy of the nasal lesions, which was processed by Pathology and
Microbiology
division in
The
laboratory received three fragments of brown
fibrous tissue, with a volume average of 1 cm3, which were
macerated
in aseptic conditions and were sown in
The
macerated Gram stain did not show microorganisms.
The post
incubation showed cream colonies growing,
round, with entire border and mucoid
aspect in solid
media, except in the McConkey Agar ( where
only grew
three lactose fermenting colonies after 24 hours incubation); Gram
Taint was
realized with Gram negative bacilli, negative oxidase,
the identification and antibiotic sensitivity test was scheduled
according to Vitek 2 methodology (System
Version 04.01); 24 hours later
was identified with a probability of 99% of microorganism like Klebsiellapneumoniae subsp. rhinoscleromatis, all antibiotic
sensitive.
The
bacteriological diagnosis was confirmed a month
later, by pathologic anatomy, whose biopsy report described pseudoepitheliomatous
hyperplasia tissue, and dense lymphocytic inflammatory infiltration in
lamina propria, plasmatic cells, Russel
Bodies and vacuolated cytoplasmmacrophages,
with
microorganisms and detritus presence. These histopathological
changes are characteristic of Rhinoscleroma,
with
Gram stain was observed Gram negative bacilli with foamy macrophages;
with
special stain, and there was no evidence of mycobacteria
or fungi.
Seven
weeks after the biopsy remained initial physical
signs and a CT scan showed the absence of maxillary sinuses lesions,
that’s why Ciprofloxacin 500mg two times a day oral via per three weeks
was initiated. Then in that time, ORL documented rhinoscleroma
diagnosis based on the bacteriological test. The patient confirmed
feeling
better, with left nasal nostril patency (LNN), right nasal nostril
(RNN)
clogged and with crusted and the dosage of the initial treatment lasted
two
more months.
After this
period the patient reported an overall
improvement in LNN, but rhinorrhea of the
RNN,
remained blocked. Treatment continued for four more months, with total rhinorrhea resolution and permanent obstruction
of RNN.
In
January, 2009, the patient claimed to have any
trouble, so that schedule was judged to scar tissue surgical removal
present in
RNN. In September 2009, as an etiological standpoint, Infectious
Diseases
Department discharged him.
Discussion
Among the
specific pathogen associated Klebsiella
infections (rare), are the rhinoscleroma
and ozena; both
diseases presents specific anato
pathologic changes. Rhinoscleroma is a
chronic granulomatous
infection located in high respiratory tract, primarily in the nasopharynx. This infection is caused by K
pneumonia subsp.
Rhinoscleromatis with is
cosmopolitan,
but endemic in some Oriental Europe zones, Latin America, Central
Africa and
South Asia and affects predominantly women. The rhinoscleroma
is acquired by direct or indirect contact with nasal discharged from a infected person and may affect bronchi, causing
hoarseness, dyspnea, stridor,
and productive cough with laryngeal tracheal engagement. The
principally
warning effect is the obstruction of air flow, which may need
endoscopic
treatment, thus progresses through three stages: a catarrhal phase
(with nonspecificinflammation),aproliferative(withonegranulomatous
aspect reaction) and a phase with the formation of scar tissue in
reparation. (See
Table 1 for an overview). Histologically,
it is
characterized by the vacuolated foamy macrophages presence, with
bacilli known
as Mikulicz cells (see Figure 1); the
determination
factors that lead their formation are unknown. The auto phagocytosis
can contribute to the fagosoma distention
and the
consequent membrane vacuole destruction, which does not allow adequate
bacilli
elimination and cause liberation into the interstitium
and implicates an immunologic macrophages deprotection.
8,11
Ozena
is a atrophic chronic rhinitis illness, characterized by nasal mucosal
necrosis
and, mucus purulentdischargers, however,
the K. pneumoniae subsp. ozenae
isolation in this study, indicates the disease, it appears to be
endemic in
subtropical and temperate regions like South Asia, Africa, Oriental
Europe and
the Mediterranean, and has been associated to patients that lives in
poorly
conditions and deficient hygiene.12
Because K.
pneumoniaesubsp.
Rhinoscleromatis is not normally found
in nasal
secretions, the culture of this type of sample is diagnostic. It has
been
routinely observed in routine practice that using McConkey
Agar media, this microorganism is recoverable only in a 50-60% of the
cases.11
The de K.
pneumoniae subsp.
Rhinoscleromatis pathogenicity
has been attributed to the composition of capsular polysaccharides,
serotype K3
specific, which protects the microorganism from phagocytosis.
The contact of illness patients with healthy persons do not transmit de
disease
in a direct way, which suggest the host susceptibility plays an
important role
in the development of the disease. In the injury sites has been
documented, a
CD4+/CD8+ lymphocytic relation altered, with a decrease of CD4+ and a
CD8+
increased, which possibility induces to alter or diminish Lymphocytes T
response.
The Rhinoscleroma Treatment suggestsTetracyclines
and Quinolones
use for a six month period or until the nasal biopsy culture were
negative;8 the
Ciprofloxacin and rifampicin combination
is interesting for the
concentrations achieved within macrophages and nasal secretions.8,11
References
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