Keywords left pulmonary vein thrombosis - adult congenital cystic adenomatoid malformation
type 2 - lower lobectomy
Introduction
Pulmonary vein thrombosis (PVT) is rarely reported after lower lobectomy.[1 ] Adult congenital cystic adenomatoid malformation Type 2 (CCAM) is rare in adults.[2 ] Here we present an unusual and interesting case of left lower lobectomy that was
performed due to CCAM, which further led to the development of PVT and hemiparesis.
Case Description
A 40-year-old female presented with complaints of fever of 6 months duration, which
was high grade and continuous, associated with cough, expectoration, and chest pain.
Initial chest X-ray ([Fig. 1 ]) was suggestive of left pleural effusion, for which patient underwent a pleural
tapping and was symptomatically better after the initiation of therapy. After 10 days,
patient started to have fever again and underwent contrast enhanced computerized tomography
(CECT) thorax ([Figs. 2 ]
[3 ]) which was suggestive of a well-defined, hypodense, and thin-walled nonenhancing
uniloculated lesion-posterior basal segment of left lower lobe indicative of intralobar
sequestration, having arterial supply to the lesion by two small branches arising
from descending thoracic aorta and venous drainage into the pulmonary circulation
([Fig. 4 ]). Bronchoscopy was done to rule out endobronchial communication of the lesion that
showed no communication, thus indicating intralobar sequestration ([Fig. 5 ]). Hence, patient underwent left lower lobectomy which was uneventful intraoperatively
and postoperatively. The patient was discharged on postoperative day 6.
Fig. 1 Chest X-ray: well-defined opacity in the left lower zone near the cardiophrenic region.
Fig. 2 Computed tomography thorax: hypodense cystic lesion in the posterior basal region
of the left lobe.
Fig. 3 Contrast enhanced computerized tomography thorax: well-defined rounded, hypodense,
thin-walled nonenhancing uniloculated lesion—posterior basal segment of the left lower
lobe. No surrounding consolidation or associated lesions.
Fig. 4 Contrast enhanced computerized tomography thorax: vascular supply—two small arterial
branches arising from descending thoracic aorta supplying the lesion and venous drainage
into the pulmonary circulation.
Fig. 5 Bronchoscopy: no endobronchial communication suggestive of intralobar sequestration.
The tissue was subjected for histological examination, and diagnosis of congenital
cystic adenomatoid malformation-type 2 was made ([Figs. 6 ]
[7 ]).
Fig. 6 Resected left lower lung lobe.
Fig. 7 yst lined by ciliated columnar epithelium having a uniform
nucleus, cyst contains serous secretions.
In total, 4 days of postdischarge, the patient again presented in the casualty with
complaints of left hemiparesis (upper and lower limb weakness). Magnetic resonance
imaging evaluation was suggestive of acute infarct in the right frontal lobe and basal
ganglia. Right middle cerebral artery thrombosis ([Fig. 8 ]). The patient was further evaluated for the cause of the cerebrovascular accident,
and on evaluation with echocardiography, it was suggestive of left PVT extending to
the left atrium.
Fig. 8 Magnetic resonance imaging brain: acute infarct in the right frontal lobe and basal
ganglia. Right middle cerebral artery thrombosis.
Treatment
The patient was started on anticoagulation therapy along with antiplatelets and with
regular physiotherapy. On follow-up after 1 month of treatment initiation, the patient
has regained partial mobility of the left upper and lower limb.
Discussion
Patients undergoing partial or complete lung lobe resection are prone to develop PVT.
Following lobectomy, the risk of PVT is 3.6%, which elevates to 13.6% in those undergoing
left upper lobectomy[3 ]
[4 ]
[5 ]. Signs and symptoms include dyspnea, hemoptysis, chest pain, fever, and hypoxemia.
In severe cases, the patient may develop a transient ischemic attack or a stroke.
In the case reported here, the patient developed PVT after left lower lobectomy which
has not yet been published in the literature to the best of our knowledge. In available
literature, there have been only six reported cases of PVT after lobectomy.[6 ] All these cases involved resection of the left upper lobe with subsequent thrombus
formation in the left upper PV stump. The true incidence of PVT after lobectomy is
unknown and likely to be underdiagnosed, especially in asymptomatic patients who do
not undergo postoperative imaging with transesophageal echo (TEE) or computerized
tomography (CT).
There is no sufficient literature evidence to suggest the routine use of postprocedure
prophylactic anticoagulation or routine imaging and echocardiography in patients undergoing
lung lower lobectomy.
CCAM is a congenital abnormality of the lung, which is uncommon in adults. Most CCAM
involves unilateral lobes of the lung and may be complicated with pulmonary bacterial
infections and abscesses. CCAM accompanied by Aspergillus infection may occur in bilateral
lobes of the lung. The clinical and radiological findings vary, thus pathological
examinations are required to make a definite diagnosis of cystic lesions of the lung.
Type 2 CCAM represents up to 25% of all congenital lung lesions, and the vast majority
are diagnosed within the first 2 years of life and occasionally present later in childhood
but is a rare entity in adulthood.
Conclusion
In conclusion, the present case has showed that thrombus in pulmonary vein stump which
is common after left upper lobectomy can still happen/occur after left lower lobectomy.
Hence, it is recommended also to have routine echocardiographic evaluation (and CECT
thorax if necessary) for patients undergoing left lower lobectomy in the same way
as it is customarily done for patients with left upper lobectomy.
If cystic lesions are found in lower lobes of lungs, CCAM should be considered in
the differential diagnosis even in adults even though they are rare.
A multicentered clinical study is advisable to elucidate the causes, frequency of
PVT and also to evaluate and validate anticoagulant therapy for PVT following left
lower lobectomy.
