Psittacosis infection and tracheobronchomalacia in a patient undergoing thyroidectomy

Article (PDF Available)inBMJ British medical journal · January 2011with24 Reads
Abstract
A 34-year-old woman with papillary thyroid carcinoma underwent total thyroidectomy with central and lateral lymphadenectomy. Immediate airway compromise required re-intubation immediately after surgery. Marked tracheal and bronchial collapse of greater than 50% of the lumen indicated tracheobronchomalacia. Subsequent attempts at extubation failed over the next week. The patient soon developed evidence of a lower respiratory tract infection. Empirical treatment with penicillins was unsuccessful. A clinical suspicion of chlamydia infection prompted initiation of macrolide treatment followed by resolution of both the patient's respiratory infection and tracheobronchomalacia. Serology returned positive for Chlamydophila psittaci infection. It later transpired that the patient had symptoms of an upper respiratory tract infection just prior to surgery. This case demonstrates an interesting and unreported cause of tracheobronchomalacia as well as providing a good lesson on the importance of preoperative screening for infection.

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BMJ Case Reports 2011; doi:10.1136/bcr.07.2011.4516
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BACKGROUND
Optimising perioperative care is paramount in any surgical
case. Pulmonary vulnerabilities and associated complica-
tions have signifi cant impact on perioperative morbidity
and mortality.
1
2
Preoperative screening for respiratory
tract infections routinely informs a deferral of elective sur-
gery. Consequently, data analysing its impact on outcome
are limited.
Psittacosis is a zoonosis caused by the obligate intracel-
lular bacterium Chlamydophila psittaci .
3
Its name pertains
to the parrot family of Psittacidae. The atypical respira-
tory tract infection is usually directly acquired from close
contact with infected birds.
4
Confi rmed cases of human
psittacosis are relatively rare. In 2008, only 61 cases were
reported in England and Wales, though this is thought
to be a marked underestimation.
5
Underlying a likely
misrepresentation is the wide spectrum of presentations
from asymptomatic to life-threatening – as defi ned by the
Centres for Disease Control (CDC) and the UK Health
Protection Agency.
4
7
Subclinical infections are usually
self-limiting. They have uncertain impact in the context
of surgery.
We report the case of a young woman who, during an
elective total thyroidectomy and modifi ed radical neck
dissection, developed an unusual life-threatening compli-
cation secondary to a co-existent initially undiagnosed res-
piratory tract infection caused by C psittaci.
CASE PRESENTATION
A 34-year-old, previously healthy woman and non-smoker,
presented with a left-sided thyroid nodule and two pal-
pable left-sided level V lymph nodes. She was clinically
euthyroid with a thyroid stimulating hormone of 1.90
(normal range 0.30–5.50 mU/l), and a free thyroxine 14.1
(9.0–25.0 pmol/l). Fine needle aspiration of the thyroid
nodule established a microfollicular pattern and histol-
ogy of a lymph node biopsy found extensive infi ltration
with a metastatic papillary thyroid carcinoma. Subsequent
CT delineated the left-sided thyroid nodule, a large left-
sided level V lymph node and a level III lymph node. Small
volume lymph nodes were also noted in the central com-
partment. Based on these fi ndings it was decided that the
patient would most benefi t from a total thyroidectomy
and bilateral modifi ed radical neck dissection with level II
to VII clearance.
On admission, 12 h prior to surgery, the patient denied
any new symptoms. Full clinical examination was equally
unremarkable and vital signs were all within normal lim-
its. Preoperative routine blood tests showed normal renal
and liver function, a C-reactive protein of less than 5
mg/l and a mild lymphocytosis of 4.3 (1.4–4.0×10
9
/l). In
theatre, the patient had an uneventful anaesthetic induc-
tion and endotracheal intubation. Total thyroidectomy
with modifi ed radical neck dissection was performed
successfully with no immediate directly associated com-
plications – recurrent laryngeal nerve intact bilaterally,
satisfactory haemostasis achieved, parathyroid glands
preserved and no tracheal resection as there was no tra-
cheal invasion.
Postoperatively on extubating the patient and on
removing positive end-expiratory pressure (PEEP) there
was acute marked oxygen desaturation requiring imme-
diate re-intubation. Emergency bronchoscopy revealed
collapse of greater than 50% of the lumen of the lower tra-
chea and large bronchi indicative of tracheobronchoma-
lacia. Subsequent attempts at reducing the PEEP were
Novel diagnostic procedure
Psittacosis infection and tracheobronchomalacia in a patient
undergoing thyroidectomy
Luke Dixon,
1
Salvador Diaz-Cano,
2
Klaus-Martin Schulte
3
1
Department of General Surgery, King’s College Hospital, King’s Health Partners, London, UK ;
2
Histopathology Department, King’s College Hospital, King’s Health Partners, London, UK ;
3
Endocrine Surgery Department, King’s College Hospital, King’s Health Partners, London, UK
Correspondence to Dr Klaus-Martin Schulte, klaus-martin.schulte@nhs.net
Summary
A 34-year-old woman with papillary thyroid carcinoma underwent total thyroidectomy with central and lateral lymphadenectomy. Immediate
airway compromise required re-intubation immediately after surgery. Marked tracheal and bronchial collapse of greater than 50% of the lumen
indicated tracheobronchomalacia. Subsequent attempts at extubation failed over the next week. The patient soon developed evidence of a
lower respiratory tract infection. Empirical treatment with penicillins was unsuccessful. A clinical suspicion of chlamydia infection prompted
initiation of macrolide treatment followed by resolution of both the patient’s respiratory infection and tracheobronchomalacia. Serology
returned positive for Chlamydophila psittaci infection. It later transpired that the patient had symptoms of an upper respiratory tract infection
just prior to surgery. This case demonstrates an interesting and unreported cause of tracheobronchomalacia as well as providing a good
lesson on the importance of preoperative screening for infection.
BMJ Case Reports 2011; doi:10.1136/bcr.07.2011.4516
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also associated with immediate lower airway collapse
and compromise. Chest radiograph after re-intubation
showed no initial evidence of intrapulmonary disease.
The patient was kept intubated on an intensive care ward
subsequently. A chest fi lm 4 days later revealed opaci-
cation of the left lower zone suspicious for consolida-
tion. These radiographic observations were accompanied
by fever and respiratory compromise with increasing
neutrophilia noted from 8.97 (2.20–6.30 10×9/l) at day 4
postoperation to 14.4 at day 7. Based on signs of infection
and an early sputum culture isolating both Staphylococcus
aureus and group A streptococcus (both sensitive to peni-
cillin) the patient was commenced on intravenous co-
amoxiclav. Despite penicillin the patients infl ammatory
markers continued to increase and further attempts to
extubate and reduce PEEP were again associated with
immediate respiratory compromise secondary to tracheal
and bronchial collapse. As the airway compromise was
persisting beyond 10 days postoperatively, the patient
underwent tracheostomy.
Further elucidation of the patient’s condition via a
dynamic CT neck and chest study showed prominent col-
lapse of the right and left main bronchi distal to the tip
of the tracheostomy tube ( gure 1 ). Also noted on CT
was patchy consolidation in both lungs, greatest in the
left lower lobe, and evidence of pulmonary hypertension
with prominence of the main pulmonary artery. As no
organisms were identifi ed with repeat blood and sputum
cultures an atypical pneumonia serology screen was per-
formed and the patient was started on macrolide therapy
to cover causes of atypical pneumonia. Complement fi xa-
tion found a signifi cant increase in the chlamydia antibody
titre and subsequent micro-immunofl uorescent antibody
assay found a specifi c, over four fold, increase in IgG to the
C psittaci strain of 1:512 (<1:64).
TREATMENT
The patient was commenced on the macrolide antibiotic
erythromycin intravenously in high standard doses.
OUTCOME AND FOLLOW-UP
On instigation of erythromycin there was a rapid improve-
ment in the patient’s clinical condition with normalisation
of white cell count and radiographic resolution of the lower
zone consolidations. Repeat dynamic CT neck and chest
study, 14 days after the fi rst CT scan and after the course
of erythromycin, showed normal airways on both inspira-
tory and expiratory phase. Clinical improvement was con-
cluded with successful weaning of ventilatory support and
eventual tracheostomy decannulation.
Post recovery, on retrospective focused questioning of
the patient, it was discovered that she kept geese and that
2 weeks prior to surgery, she had undertaken cleaning of
her goose enclosure. The patient also reported that 2 days
before admission she had developed a sore throat, which
from fear of delaying her surgery the patient had chosen
not to disclose.
DISCUSSION
This case illustrates an unusual complication of elective
surgery and an unfamiliar presentation of C psittaci infec-
tion. It is our supposition that the patient contracted the
infection through inhalation of aerosolised organism from
cleaning her bird coop.
8
With subsequent intubation and
ventilation, the psittacosis was likely spread throughout
the respiratory tract. We propose that the C psittaci infec-
tion, disseminated to the trachea and bronchus, then led to
dispersed infl ammatory infi ltration, oedema and localised
necrosis which in turn undermined the integral strength of
the tracheal wall. This infl ammatory associated weaken-
ing could have been further compounded by the trauma
of intubation, local surgery and perhaps oxygen toxicity.
These combined factors ultimately resulting in the diffuse
tracheobronchomalacia are seen on extubation. Underlying
this suggested pathophysiology is therefore both a rare
clinical picture of C psittaci infection and a poorly charac-
terised cause of tracheobronchomalacia (based on a journal
search performed February 2011 see appendix 1).
Tracheobronchomalacia (TBM) is a disease of the central
airway in which weakness of the tracheal and bronchial
walls develop due to damage to the supporting cartilage,
predisposing to airway collapse on exhalation.
9
11
In
adults, common acquired causes include prolonged intu-
bation, severe emphysematous disease, chronic infl am-
mation and recurrent respiratory infections.
12
14
Refuting
the argument that intubation alone may have caused the
above cases TBM, is the short duration of intubation
(3–4 h) and that there was observed diffuse tracheal and
Figure 1 CT chest and neck study during expiration. Note the greater than 50% collapse of both the trachea (a) and the bronchi (b).
BMJ Case Reports 2011; doi:10.1136/bcr.07.2011.4516
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bronchial weakness, whereas typically, intubation associ-
ated TBM is characterised by a segmental weakness from
localised pressure necrosis from the cuff.
12
14
Another
possible confounding aetiology is that there are several
reported cases of TBM secondary to goitres. These cases
are however, generally associated with large compres-
sive goitres and/or tracheal invasion, neither of which
were a feature of this case.
15
19
Furthermore in a recent
review of 1969 patients with large retrosternal goitres,
there were few reported cases of postoperative tracheal
collapse.
20
We also put forth that goitre associated mala-
cia would be localised to the area of the trachea which
was in direct apposition to the thyroid, unlike the weak-
ness noted in this case which extended into the bronchi.
Related to this is the argument that the process of thy-
roidectomy in itself could lead to local infl ammation and
malacia. The generalised nature of the TBM again refutes
this as the sole cause in this case, additionally a retro-
spective review of thyroidectomy suggested minimal
risk of TBM with modern thyroid surgery.
21
In contrast,
supporting a primary infectious aetiology, is the observa-
tion that recurrent lower airways infection, and therapy-
resistant, irreversible, and/or atypical asthma, as well as
regional infl ammatory diseases (oesophageal or aortic)
are well-characterised factors associated with a diffuse
airway malacia.
22
23
With the evidence that TBM is rare
in adults without chronic respiratory conditions it could
also be argued that this patient may have had an unusual
genetic predisposition to TBM. We feel however that this
is unlikely in a patient who had no signifi cant respira-
tory medical history and that the unusual combination of
infection, intubation and surgery is enough to rationalise
the atypical presentation.
Looking at C psittaci and extending into the Chlamydia
genus there are no previous documented cases of these
infections leading to TBM (based on a journal search per-
formed February 2011 see appendix 1). Nevertheless we
feel that the patient had a concomitant psittacosis, which
complicated her surgery. Tallying with this is the observed
approximate 10 days incubation period between exposure
and infection, and the radiographic fi ndings of lower zone
consolidation both of which are consistent with recent
psittacosis.
4
24
Moreover the serological evidence, at 15
days into infection, of an observed over fourfold increase
in IgG antibodies to C psittaci on micro-immunofl uorescent
antibody assay is consistent with a probable diagnosis of
psittacosis, as classifi ed by the CDC.
25
Finally, supporting
an infectious aetiology is the dramatic clinical improve-
ment and resolution of the airway collapse post macrolide
therapy, a well-recognised treatment of psittacosis.
26
27
In closing, although this case highlights a life-threatening
complication of surgery secondary to psittacosis, specifi c
routine preoperative screening for C psittaci is arguably not
justifi able due to the supposed rarity of this presentation
and possible resource limitations. Nevertheless this case
heavily highlights the essential need for a detailed clinical
examination and history prior to surgery. It is our belief
that if, in this patient’s case, the pre-existent respiratory
tract infection had been diagnosed and empirically treated,
with an appropriate delay in surgery, tracheobronchoma-
lacia and the associated high morbidity would have been
avoided.
Learning points
On admission for surgery a thorough clinical
examination and history is essential in all cases to
reduce the risk of major complications.
If a preoperative concomitant infection is diagnosed
then surgery should be delayed or at the very least
related complications expectantly managed.
C psittaci is widely thought to be an under diagnosed
infection, clinicians should be aware of it as it is often
readily treatable.
Even minor respiratory tract infections can be life-
threatening in the context of major surgery.
In cases of tracheobronchomalacia an infectious
aetiology, such as psittacosis, should be considered.
Competing interests None.
Patient consent Obtained.
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APPENDIX
1. On the 10th of June 2001 a journal search for the terms – tracheobronchomalacia/ tracheomalacia/ tracheitis and chlamydia, tracheobronchomalacia/ tracheomalacia/
tracheitis and chlamydophila, tracheobronchomalacia/ tracheomalacia/ tracheitis and psittacosis – in Pubmed, Cochrane library, EMBASE, MEDLINE, BMJ Case
reports returned 0 relevant results.
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