Mycobacterium fortuitum-induced persistent parotitis: successful therapy with clarithromycin and ciprofloxacin.

Abstract

Parotitis caused by nontuberculous mycobacteria, a very rare disease entity, has never been reported to be caused by Mycobacterium fortuitum (M. fortuitum) in the literature.
An 8-year-old girl was seen with painful swelling of the right parotid gland despite antibiotic treatment of more than 1 month. Elevated serum amylase activity and diffuse contrast-enhanced CT of the parotid gland confirmed the diagnosis of parotitis. Histopathological study of specimens taken from the right parotid tail mass showed granulomatous inflammation with acid-fast positive bacilli; culture later confirmed M. fortuitum. After administration of clarithromycin and ciprofloxacin for 9 consecutive months, the parotitis and parotid tail mass were completely resolved at follow-up examination.
To our knowledge, this is the first case report of parotitis caused by M. fortuitum and its successful medical treatment.

Full text

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CASE REPORT
Dennis H. Kraus, MD, Section Editor
MYCOBACTERIUM FORTUITUM–INDUCED PERSISTENT
PAROTITIS: SUCCESSFUL THERAPY WITH
CLARITHROMYCIN AND CIPROFLOXACIN
Chien-Cheng Chen, MD,1Shiou-Yi Chen, MD,1Yun-Sung Chen, MD,2
Cheng-Yu Lo, MD,3Po-Wen Cheng, MD1
1Department of Otolaryngology, Far Eastern Memorial Hospital, Taipei, Taiwan. E-mail: powenjapan@
yahoo.com.tw
2Department of Internal Medicine, Division of Pulmonary Medicine, Far Eastern Memorial Hospital,
Taipei, Taiwan
3Department of Pathology, Far Eastern Memorial Hospital, Taipei, Taiwan
Accepted 19 December 2006
Published online 11 April 2007 in Wiley InterScience (www.interscience.wiley.com). DOI: 10.1002/hed.20626
Abstract:
mycobacteria, a very rare disease entity, has never been
reported to be caused by Mycobacterium fortuitum (M. fortui-
tum) in the literature.
Methods and Results. An 8-year-old girl was seen with pain-
ful swelling of the right parotid gland despite antibiotic treatment
of more than 1 month. Elevated serum amylase activity and dif-
fuse contrast-enhanced CT of the parotid gland confirmed the
diagnosis of parotitis. Histopathological study of specimens
taken from the right parotid tail mass showed granulomatous
inflammation with acid-fast positive bacilli; culture later con-
firmed M. fortuitum. After administration of clarithromycin and
ciprofloxacin for 9 consecutive months, the parotitis and parotid
tail mass were completely resolved at follow-up examination.
Conclusion. To our knowledge, this is the first case report of
parotitis caused by M. fortuitum and its successful medical treat-
ment.
V V
1064, 2007
Background. Parotitis caused by nontuberculous
C2007 Wiley Periodicals, Inc. Head Neck 29: 1061–
Keywords: Mycobacterium fortuitum; nontuberculous mycobac-
teria; parotitis; clarithromycin; ciprofloxacin
Primary salivary gland tuberculosis is common,
occurring more frequently in the parotid gland
than in other salivary glands. Compared with
Mycobacterium tuberculosis, primary nontuber-
culous mycobacterial infection of the parotid
gland is a very rare disease, usually manifested
unilaterally as either an acute inflammatory pro-
cess or a chronic tumorous lesion.1The acute
inflammatory lesion poses a challenge to clini-
cians, because it mimics acute bacterial parotitis
or mumps.
Mycobacterium fortuitum (M. fortuitum), a
nontuberculous mycobacteria (NTM), is generally
accepted as a skin commensal in humans.2Al-
though cervical lymphadenitis caused by M. fortu-
itum has been reported in the literature,3,4paroti-
tis caused by M. fortuitum has never been
reported. Recently, we experienced an 8-year-old
girl with persistent parotitis and parotid tail mass
attributed to the infection by M. fortuitum.
Herein, we present thiscase.
Correspondence to: P.-W. Cheng
V V
C2007 Wiley Periodicals, Inc.
Mycobacterium fortuitum–Induced ParotitisHEAD & NECK—DOI 10.1002/hedNovember 2007 1061

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CASE REPORT
An 8-year-old girl had persistent painful swelling
of the right preauricular area for more than
1 month. She was 27 kg in weight and 130 cm in
height. She denied fever, generalized malaise,
weight loss, cough, or sputum. She also denied
history of oral trauma or dental procedures. Her
medical and past histories were unremarkable,
and all vaccinations (including measles-mumps-
rubella [MMR] vaccine) were on schedule. Ini-
tially, she had been diagnosed for acute suppura-
tive parotitis, but oral treatment with 10 mL Aug-
mentin syrup (each 5 mL containing 125 mg
amoxicillin and 31.25 mg clavulanic acid) every
8 hours for 2 weeks showed no improvement. At
admission, physical examination revealed gener-
alized tender swelling of the right parotid gland
with a 3.0- 3 3.0-cm mass at the parotid tail.
Laboratory tests revealed elevated serum amy-
lase activity (156 IU/L; normal range, 30–110 IU/
L) and leukocytosis (13,420 cells/lL). The result
for HIVantibody was negative. There was no pres-
ence of a 4-fold rise in serum mumps immunoglob-
ulin G titer between acute and convalescent
phase. A CT scan of the head and neck demon-
strated diffuse enhancement of the right parotid
gland (Figure 1) as well as a 3.3- 3 3.0- 3 2.3-cm
soft tissue mass at the parotid tail, which showed
central low-density with peripheral rim enhance-
ment (Figure 2). Additionally, CT scan ruled out
cervical lymph node involvement. Histopatho-
logy of biopsy specimens from the parotid tail
mass revealed granulomatous inflammation (see
Figure 3) with acid-fast positive bacilli (Figure 4).
At first, primary M. tuberculosis of the parotid
gland was suspected, and the patient was treated
with ethambutol (400 mg once a day), pyrazi-
namide (1000 mg once a day), and Rifinah-300
FIGURE 1. Axial contrast-enhanced CT scan shows diffuse
enhancement of the right parotid gland (arrows) compared with
the left side.
FIGURE 2. Contrast-enhanced axial CT image demonstrates
asymmetrical right parotid tail mass (arrows) associated with
low-density ring enhancement.
FIGURE 3. The tissue section of the parotid tail mass stained
withhematoxylin-eosinshows
(arrows) with lymphoplasmacytic infiltrate and focal necrosis.
Foci of histiocyte aggregate and a few multinucleate giant cells
are present. [Color figure can be viewed in the online issue,
which is available at www.interscience.wiley.com.]
granulmatousinflammation
1062Mycobacterium fortuitum–Induced ParotitisHEAD & NECK—DOI 10.1002/hedNovember 2007

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(300 mg rifampicin and 150 mg isoniazid; once a
day). Unfortunately, persistent parotitis and poor
wound healing were noticed. Two months later,
acid-fast bacilli culture grew M. fortuitum. Drug
susceptibility report showed the organism to be
sensitive to imipenem, amikacin, cefoxitin, cipro-
floxacin, and clarithromycin. Subsequently, cipro-
floxacin (125 mg twice a day) and clarithromycin
(250 mg twice a day) were given for 9 consecutive
months. She has done well, and her 1-year follow
up revealed no subsequent problems.
DISCUSSION
The Runyon Classification divided NTM into 4
groups based on colonial morphology, pigmenta-
tion, and growth rate.4Of the groups, M. fortui-
tum is classified as Runyon group IV, rapidly
growing mycobacteria. Rapid growth is character-
istic only for subcultured organisms which form
well-developed colonies after just 7 days ofincuba-
tion, while their primary isolation may take 3 to 6
weeks.5
M. fortuitum has been isolated from soil, dust,
water sources (including hospital sinks), human
abscess, and sputum,2but no strong evidence sup-
ports person-to-person spread.6However, M. for-
tuitum can infect almost every tissue and organ
system. It has been implicated in skin and soft
tissue infections, trauma site and catheter-
related infections,7keratitis,8peritonitis, artifi-
cial heart valve replacement infections, augmen-
tation mammoplasty, cardiothoracic surgery, and
arthroplasty.9In general, M. fortuitum usually
causes localized infections with low mortality in
immunocompetent patients. Disseminated dis-
ease, usually with skin and soft tissue lesions,
occurs almost exclusively in the setting of severe
immunosuppresion, especially acquired immuno-
deficiency syndrome.3,4,9Deep organ involvement
and disseminated infections manifest in highmor-
tality.9
Most patients with cervical lymphadenitis
caused by NTM infection are children. They usu-
ally show normal chest radiograph and have no
fever, malaise, or weight loss.10,11
enhanced CT scan typically demonstrates asym-
metrical cervical lymphadenitis and contiguous
low-density, necrotic, ring-enhancing masses but
with minimal inflammatory stranding of the sub-
cutaneous fat, a finding that may distinguish
lymphadenitis caused by NTM from other conven-
tional bacteria.12,13Among NTM, M. fortuitum is
an extremely rare cause of isolated lymphadenitis
or neck abscess.4,10,11,13A history of dental proce-
dures or defects in the immune system such as
human immunodeficiency virus infection may
provide insight to the cause of such an infection.4
However, in the present case, she was neither an
immunocompromised patient nor receiving any
dental procedure; CT scan ruled out cervical
lymphadenitis.
NTM-induced parotitis is very rare, and to our
knowledge, M. fortuitum-induced parotitis has
never been reported in the literature. One of the
main etiologic factors that may facilitate parotitis
is decreased salivary production. Common causes
of decreased salivary production include chemo-
therapy and radiotherapy, autoimmune disease
and/or xerogenic medication. The patient, how-
ever, exhibited none of the above chararcteristics.
Thus, we inferred that reduced saliva production
due to ductal obstruction might be the predispos-
ing factor for M. fortuitum-induced parotitis by
means of an ascending salivary duct infection via
the oral cavity.
Guidelines for treatingpatientsspecificallywith
parotitis caused by M. fortuitum are lacking in the
literature. In the past, NTM generally have shown
poor in vitro susceptibility to standard antitubercu-
lous drugs,14and many authors have recommended
surgical interventions including partial or total
parotidectomies for NTM infection of the parotid
gland lymph nodes.13,15,16Conversely, many studies
show that clarithromycin, amikacin, cefoxitin, fluo-
roquinolones (ciprofloxacin and ofloxacin), doxycy-
cline, imipenem, and sulfonamides have good in
vitro activity against M. fortuitum.17,18A combina-
Contrast-
FIGURE 4. The Kinyoun acid-fast stained histopathology sec-
tion of the parotid tail mass displays rod-shaped bacteria
(arrows). [Color figure can be viewed in the online issue, which
is available at www.interscience.wiley.com.]
Mycobacterium fortuitum–Induced ParotitisHEAD & NECK—DOI 10.1002/hedNovember2007 1063

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tionof2ormoreagentsshouldalwaysbeutilized,as
with all mycobacterial infections, to avoid drug re-
sistance. Based on the present case and invitro data
in the literature, our recommendation is to treat M.
fortuitum-induced parotitis with a combination of
ciprofloxacin and clarithromycin for at least 9
months.
Acknowledgment.
for manuscript editing.
We thank Anthony Lee
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