ARTICLE
Auteur(s) : Mar
Llamas-Velasco1, Inmaculada Domínguez1,
Enrique Ovejero2, Silvia Pérez-Gala1, Amaro
García-Diez1
1Department of Dermatology, Hospital
Universitario de la Princesa, C/ Diego de León 62, CP 28006,
Madrid, Spain
2Department of Thoracic Surgery, Hospital
Universitario de la Princesa, C/ Diego de León 62, CP 28006,
Madrid, Spain
accepté le 26 Août 2009
Empyema necessitatis (EN) is defined as the extension of
purulent pleural liquid through adjacent tissues to form an abscess
on the thoracic wall. It was first described in 1640 by Baillon [1]
and subsequently by Laennec [2] in 1890. Cutaneous manifestations
of EN are unspecific. The most important one is the presence of a
subacute mass on the costal wall. Thus, dermatologists should be
aware of this disease because sometimes they are the first
physician to attend these patients.
Case report
A 32-year-old woman went to her primary care physician with a
one-month history of non-productive coughing and an enlarging,
painful mass on the anterior right thoracic wall. There were no
symptoms of fever, dyspnea, thoracic pain or any other. She was
diagnosed with nervous anorexia eight years ago and also had a
laxative abuse history. The patient was sent to hospital with an
altered chest X-ray showing a shadow on the right lung, along with
ipsilateral pleural effusion. Physical examination showed
moderate-illness with generalised wasting of muscle and
subcutaneous fat. She was found to have a pink erythematous, warm,
tender mass of 7 centimetres in diameter, between the
5th and 10th ribs, with moderate oedema and a
fluctuant centre (figure
1). Poor oral hygiene with several caries was observed.
Laboratory values revealed a white blood-cell count of
10,240/mm3 with 91.5% neutrophils. Hemoglobin was
11.1 g/dL and platelets were 368,000/mm3. Kidney,
liver and thyroid tests were all within normal limits. Several
serologies, (Chlamydia, Q-fever, Adenovirus, Mycoplasma,
Legionella, Respiratory Syncytial Virus and HIV), were negative.
She had a compensated metabolic alkalosis that was thought to be a
secondary effect of the laxative abuse. Purified protein derivative
(PPD) for a tuberculous skin test was non-reactive. Intravenous
cotrimoxazole (400 mg/12 h) and intravenous imipenem
(500 mg/8 h) were prescribed at this moment as empiric
therapy for fifteen days. A computed tomography scan of the
chest revealed a small-sized right pleural effusion and pulmonary
infiltration without demonstration of a strange endobronchial body
(figure 2). It
also revealed the presence of a continuity solution between the
pleural empyema and the adjacent subcutaneous tissue. Because of
these findings, a fine-needle aspiration biopsy (FNAB) of the mass
was performed for microbiological and cytological study.
Afterwards, the patient was sent to Thoracic Surgery for drainage
of purulent pleural collections. Cytological studies were not
useful. The microbiological studies revealed Gram-positive ramified
bacillus. Culture results were positive for Actinomyces
gerencseriae, Klebsiella corrodens and negative-coagulase
Streptococci. Fungal and mycobacterial smears and cultures were
negative. Thus, Actinomyces gerencseriae was thought to be the
etiological agent of her disease, and she was diagnosed with
empyema necessitatis secondary to thoracic actinomycosis (TA).
Specific antibiotic treatment with intravenous amoxicillin plus
clavulanic acid (2 g/8 h doses, for fifteen days), and
oral amoxicillin plus clavulanic acid (875 mg/8 h, for
two and a half months) were used, resulting in lung improvement and
important clinical progress.
Discussion
EN is most commonly reported in adults. We could not find
prevalence and incidence data about EN, although global incidence
is thought to be decreasing. There are non-infectious causes of EN
such as carcinoma, mesothelioma and lymphoma [3, 4], but infectious
ones are the most important. Tuberculosis, actinomycosis, and
pyogenic bacteria are the most commonly reported, but fungi are
also found [5-7]. This rare complication of pleural space infection
occurs mostly due to inadequate or late treatment of such
infections. Actinomyces is a gram-positive, non-spore forming,
predominantly anaerobic prokaryotic bacteria. Most of EN caused by
actinomycetes are produced by A. israelii but A. odontolyticus has
also been described as a causal agent [8]. A. gerencseriae, the
etiological bacteria in our case, is considered the serovariety 2
of A. Israelii [9]. Recently, Freeman et al. reviewed 26
cases of EN in the English medical literature from 1966 to 2004
[5]. They found that 50% were the result of M. tuberculosis and 24%
were due to Actinomyces infection. A computerized search for
reports in the English and Spanish literature up to 2004 was
carried out using Pubmed and Medline and a summary of those cases
of EN, excluding non-infectious causes, can be seen in table 1. We found 18 articles, including ours,
describing 20 cases (table 1 [10-17]).
Only one article included several cases [7]. Most of the patients
included (n = 15, 75%) were men. Patients were aged from 3
months-old to 88-year-old. Mycobacterium tuberculosis was less
frequent than in previous reports (n = 7, 35%) but it is still
necessary to rule out this etiology. Instead, thoracic
actinomycosis seems to be slightly more relevant nowadays (n = 5,
25%). The remaining cases (n = 8, 40%) were the result of
infections by S. aureus, E. coli and Aspergillus spp. Therefore,
there has been a change in the causal agents. It is remarkable that
the etiological agent is methicillin resistant
Staphylococcus aureus (MRSA) in two cases, both of them in USA
reports [18, 19]. This fact can be a sign of the increasing
importance of this pathogen in several countries, especially in the
USA.
In empyema development, pleural fluid passes through three
pathogenic stages of increasing host defence activity and bacterial
invasion [20, 21]. In the first one, which is also called the
exudative stage, bacterial infection and inflammation release
cytokines (IL-8, TNF-α) that increase tissue and capillary
permeability. In the second one, called the fibrinopurulent stage,
pleural fluid is invaded by bacterium. In the last stage there is a
fibroblastic proliferation in the inner surface of the pleura. This
whole process is known to be driven by mediators such as
transforming growth factor (TGF-β) and platelet derived
growth-factor (PDGF) [22].
Clinically, patients mostly had an enlarging, painful, slightly
erythematous, moderate demarcated mass, in the chest wall (n = 8,
40%), cough (n = 6, 30%), pleuritic-type chest pain (n = 4, 20%)
and dyspnea (n = 2, 10%). Although a few cases with mastitis-like
debut have been described [6], none of these reported patients
presented it. Extension tends to occur along the pathway of least
resistance and in this manner, EN usually affects between the
second and sixth intercostal spaces [23] as in our case. Three
cases (cases 5, 6 and ours) had poor teeth care [24, 25] but no
one, apart from our patient, had anorexia. Symptoms for TA are
unspecific. Constitutional symptoms usually begin between two
months and six years prior to disease characterization, which has a
medium delay of six months [26, 27]. Thus, delayed diagnosis or
misdiagnosis such as tuberculosis, lung abscess or lung tumour is
common and inadequate surgical approaches can be taken [28].
Sometimes cutaneous signs such as chest wall swelling, appearance
of an abscess or thoracic skin fistulation are present from the
beginning [29] and we can obtain an earlier diagnosis. We find risk
factors such as chronic alcoholism, caquexia, EPOC, TBC, poor teeth
health, bronchiectasias, etc., in 50% of patients with TA. There
are also several cases associated with intrabronchial bodies [30],
tumours or even concomitant tuberculous disease, but there is no
higher incidence in HIV patients [29]. The finding of a lithic
lesion on the ribs in X-rays is highly suspicious but infrequent
[31].
EN diagnosis requires demonstration of continuity between the
purulent pleural fluid and the thoracic wall abscess, so we usually
need TC to diagnose. FNAB (fine-needle aspiration biopsy) is a very
useful diagnosis method in EN [7, 32]. This technique allows us to
make a cytological diagnosis as well as to obtain material for
culture. EN has rarely been reported to extend to abdominal spaces
or to the head and neck [33-35] or in the reverse pathway [24]. On
the other hand, actinomycosis diagnosis requires the demonstration,
by culture, of the etiological agent but sputum culture is not
useful because Actinomyces genre is a common oropharynx inhabitant
[31]. Blood culture is usually negative, but there are several
cases with septic dissemination [36]. Various bacteria have all
been isolated, with Actinomyces spp in some combinations [37]. In
our case, we found Klebsiella corrodens and negative-coagulase
Streptococci, both previously described. They are thought to
enhance the pathogenicity of Actinomyces by creating an anaerobic
environment.
In the pre-antibiotic era, mortality was globally 66% [27]. The
mortality rate is nowadays less than 5% in infectious cases because
most of them respond promptly to antibiotics. In contrast,
neoplastic EN portends a poor prognosis with limited survival [7].
EN mortality mechanisms are all interconnected and can co-exist.
They can be grouped as follows: 1) respiratory insufficiency, with
diminished lung or chest wall elasticity and ventilation-perfusion
matching alterations, because of intra-alveolar filling; 2) cardiac
failure, secondary to pericarditis or hypoxemia; 3) mediastinitis,
which alters proper cardiac function and can compress airways; 4)
haematogenous dissemination, that can produce hemodynamic
instability and sepsis, with multiple-organ failure; 5) acute renal
insufficiency due to dehydration and low-output cardiac failure;
and 6) iatrogenic causes such as incorrect chest-tube placement
technique or haemorrhagic problems due to the use of intrapleural
fibrinolytics. All these facts can be aggravated because of
previous diseases, and they are much more likely to happen in older
patients. EN treatment includes a double approach: medical and
surgical. Empirical antibiotic treatment (highly recommended
because decreasing morbidity and mortality rates) using a
second-generation cephalosporin or aminopenicillin with a
β-lactamase inhibitor plus anaerobic coverage or, for sensitive
patients, a combination of ciprofloxacin plus clindamycin, covers
all likely pathogens. There exist no direct data on how long to
treat empyema, but most authors advise one week with intravenous
treatment followed by 1-3 weeks of oral treatment. For actinomyces
there are some articles suggesting that shorter treatments are at
least equally effective [38] and recommending using antibiotics
whose activity fights concomitant bacteria, instead of penicillin
[39]. In our case, using this new treatment approach was very
successful. There is an expert consensus that pus within the
pleural space requires prompt tube drainage. Optimal timing of
drainage is also a controversial theme. In any case, clinical
response is the most important factor. There are also several
surgical techniques that can be used here, such as drainage with
thoracotomy, decortication and wide surgical drainage. New
techniques, such as VATS (Video-Assisted Thoracic surgery) [40],
will probably be used in the near future for this disease.
Table 1 Clinical, etiologic agents and received
treatments summary
|
Reference
|
Age/Genre
|
Predisposing
|
Clinic
|
Etiology
|
Diagnosis
|
Treatment
|
Surgery
|
|
Porcel JM [7]
|
88/M
|
Old pleural TB
|
Dyspnea+mass
|
MT
|
FNBA
|
None
|
Chest tube
|
|
Chaiyasate K [10]
|
18/M
|
None
|
Costal mass
|
MT
|
Culture
|
Anti TB
|
Unknown
|
|
Soto-Hurtado E [6]
|
65/M
|
Alcoholic. Smoker. Previous TB
|
Dyspnea+pain+constitutional syndrome
|
Assp
|
Biopsy
|
Anfotericine
|
Thoracotomy+costal resection
|
|
Pinarli FG [11]
|
9/F
|
None
|
Pleuritic pain+ anorexia
|
Asp
|
Biopsy
|
Penicillin
|
Unknown
|
|
Huits RM [25]
|
43/M
|
Caries, undernourishment
|
Cough+pleuritic pain
|
Asp
|
FNBA
|
Unknown
|
Unknown
|
|
De Wit M [24]
|
44/M
|
Caries, alcohol, colonic adenoma
|
Costal mass+non productive cough
|
EC+SA
|
Culture
|
Wide spectrum antibiotics
|
Thoraco-centesis
|
|
Moore FO [12]
|
3m/F
|
None
|
Fever + rash
|
MRSA
|
Culture
|
Vancomycin+linezolid
|
Decortication +chest tube
|
|
Fatureto M [13]
|
26/M
|
None
|
Costal mass
|
Asp
|
Biopsy
|
Ciprofloxacin
|
None
|
|
Kono SA [26]
|
52/M
|
Alcoholic, marihuana smoking
|
Cough+dyspnea+fever
|
Asp
|
UK
|
Piperacillin-tazobactam
|
VATS
|
|
Reyes CV [4]
|
68/M;79/M;29/M 47/M
|
Unknown
|
Unknown
|
MT AI
|
FNBA
|
Anti TB Penicillin
|
Pleurodesis
|
|
Tonna I [14]
|
53/M
|
None
|
Myalgia+mass
|
SA
|
Culture
|
Intravenous antibiotics
|
Surgical debridement
|
|
Lee JK [15]
|
12/F
|
None
|
Cough+constitutional syndrome
|
ASP
|
Culture
|
Penicillin+ doxycycline
|
None
|
|
Tezel C [16]
|
68/F
|
None
|
Costal mass. Pleural thickenning
|
UK
|
Culture
|
Anti TB
|
Chest tube + thoracostomy
|
|
Sennent C, et al. [18]
|
24/M
|
None
|
Fever+cough+abdominal wall mass
|
MT
|
PCR
|
Anti TB
|
Chest tube
|
|
García-Gasalla M, et al. [19]
|
50/M
|
Old pleural TB
|
Thoracic ulceration
|
MT
|
Culture
|
Anti TB
|
None
|
|
Mizell KN [17]
|
59/M
|
None
|
Costal mass+pain
|
MRSA
|
Culture
|
Vancomycin+ciprofloxacin+TMT-SMX
|
Wedge resection of upper left lobe
|
|
Llamas M, et al.
|
32/F
|
Caries, undernourishment
|
Costal mass+non productive cough
|
AG
|
FNBA
|
Amoxicyllin+clavulanic
|
Chest tube
|
Conclusion
From a dermatological point of view, we must remember that any
enlarging and painful subacute thoracic mass with fluctuation
should be considered as an EN suspicious lesion. When we suspect
it, the diagnostic approach must include a chest X-ray, to rule out
lung affectation. In that case, TC is useful to demonstrate the
presence of continuity between empyema and the subcutaneous
abscess, and, so, to consolidate the diagnosis. In any case, timely
pleural fluid analysis, when we find pleural effusion, can prevent
this complication [30]. Thus, FNAB should be considered as a first
approach to diagnosis because of the possibility of reaching an
etiologic diagnostic, which lets us choosing the most appropriate
therapy [41]. Early diagnosis and treatment reduces complications,
and that’s the reason why dermatologists should be aware of this
entity, to promote an earlier recognition and treatment.
Acknowledgements
Financial support: none. Conflict of interest: none.
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