Case
A 49-year-old woman received a routine medical check-up. An abnormal shadow was found
on chest X-ray (CXR). She visited her home doctor and underwent a CT scan. CT showed
a nodular lesion in the left lower lobe and she was referred to our hospital.
Her medical interview revealed that she suffered flu-like symptoms and hemoptysis two
months previously and was cured by medication within one week. The relation between
the symptoms and menstrual period was not clear.
Her past surgical history was an operation for descensus uteri 16 years previously
and her obstetric history was two deliveries and two abortions. Her smoking history
was 20 pack-years and she was an ex-smoker and who had quit smoking about one year
previously. Complete blood count and blood chemistry were normal. No coagulation
disorder was found.
CXR revealed a nodular shadow in the left lower lung field ( Fig. 1a and b ). CT showed a round-shaped
nodule with a smooth border in the left lower lobe adjacent to the pleura. The long
axis of the nodule was 20 mm on CT ( Fig. 2a ). The differential diagnosis was pulmonary hamartoma, sclerosing
pneumocytoma, and solitary fibrous tumor of the pleura. She was followed up by her
home doctor.
(a, b) CXR showed a nodular shadow in the left lower lung field (white arrow)
on first visiting to our hospital.
Pre- and post-contrast-enhanced CT. (a) CT showed a round-shaped nodule in
the left lower lobe on the first visit to our hospital. The nodule was
enlarged ten months later and contained (b) small high-density areas with
(c) inhomogeneous enhancement and there was no GGO or consolidation
surrounding the nodule (d).
Ten months later, hemoptysis recurred and she visited our hospital again. CT revealed
that the nodule enlarged to 30 mm in the long axis. The nodule consisted of a
low-density area and a small high-density area and showed inhomogeneous enhancement.
There was no GGO or consolidation surrounding lung parenchyma ( Fig. 2b–d ). T1-weighted (T1W) imaging and
T2-weighted (T2W) imaging both showed high and low signal intensities in the nodule;
it was partly enhanced after injection of contrast medium ( Fig. 3 ). A signal void in the nodule may have
reflected cavity formation or hemosiderin deposition. Diffusion-weighted imaging
(DWI) showed a diffusion-restricted area and an enhanced area in the nodule ( Fig. 4 ). The nodule had
increased in size and malignancy was suspected. Video-assisted thoracotomy was
performed. In thoracoscopic findings, there was no pleural lesion and the nodule was
located in the subpleural lung parenchyma. She had a good course and without any
complications after surgery.
T1W imaging, T2W imaging, and contrast-enhanced T1W imaging. T1W (a) and T2W
imaging (b) both showed high and low signal intensity areas and a small
signal void in the nodule. (c) After administration of contrast medium,
there was a nodular enhancing area in T1W imaging
(a, b) DWI and apparent diffusion coefficient (ADC) map. In DWI (b = 1000),
the nodule had a diffusion-restricted area (black arrow head) and an
enhanced area (white arrow).
Formalin-fixed material showed a yellowish-white colored nodule protruding from the
lung parenchyma. Hemorrhage was visible in the central area in the nodule.
Microscopically, endometrial tissue with many glands and interstitia were found.
There was hemorrhage with tissue laceration and hemosiderin deposition in only a
small part. In adjacent lung parenchyma, collagen was deposited in a band-like area
containing bronchiolar or other lung tissue. A large number of hemosiderin-laden
macrophages were observed in the surrounding lung parenchyma caused by pulmonary
hemorrhage ( Fig. 5 ). The
final pathological diagnosis was bronchopulmonary endometriosis.
Microscopic findings (hematoxylin and eosin stain 20). Microscopic view
revealed endometrial tissue with hemorrhage and hemosiderin-laden
macrophages surrounding the alveolar space Microscopic view revealed
endometrial tissue with hemorrhage and hemosiderin-laden macrophages
surrounding the alveolar space.
After surgery, her symptoms disappeared and no recurrence was seen even without
hormonal therapy for about five years.
Intro
Intrathoracic endometriosis is classified into pleurodiaphragmatic endometriosis and
bronchopulmonary endometriosis according to the migration of endometrial tissue.
Bronchopulmonary endometriosis is rare. Intrathoracic endometriosis frequently
occurs in the right thorax ( 1 ). Computed tomography (CT) findings of bronchopulmonary endometriosis
are lung nodules, with or without cavities, or surrounding ground-glass opacities
(GGO). Features vary with menstrual status ( 2 , 3 ).
Recently, the usefulness of magnetic resonance imaging (MRI) was reported for the
diagnosis of intrathoracic endometriosis, but most published reports were about
pleurodiaphragmatic endometriosis ( 4 – 9 ). We present CT and MRI findings of
bronchopulmonary endometriosis that showed a gradually enlarging nodule in the left
lung.
Discussion
Intrathoracic endometriosis is classified into two categories, pleurodiaphragmatic
and bronchopulmonary endometriosis. This case was categorized as the latter and
endometrial tissue was in the bronchiole or lung parenchyma. Pregnancy, delivery,
and hysterectomy are risk factors. The right thorax is predominant and hemoptysis or
hemosputum within the menstrual period are general symptoms ( 1 ). However, in other reports there was no
laterality in bronchopulmonary endometriosis compared with pleuro-diaphragmatic
endometriosis ( 2 ). Several
etiologies were hypothesized and microembolism of endometrial tissue is the most
likely one. Risk factors are trauma or uterine manipulation. According to this
hypothesis bronchopulmonary endometriosis would occur in both lungs. However,
catamenial hemoptysis is also a symptom of bronchopulmonary endometriosis, and this
symptom shows right-side predominance ( 1 ). Clearly explaining the reason for
laterality of bronchopulmonary endometriosis has not been possible and further
discussion is needed.
Bronchopulmonary endometriosis usually results in a hematoma or alveolar hemorrhage
in the lung. Consolidation, nodular shadow, or miliary shadow were reported on CXR.
CT showed consolidation, GGO, nodules with cavitation, or GGO surrounding lung
parenchyma. The lesions appeared or disappeared in relation to menstruation, so a
normal image finding was also possible ( 2 , 3 ).
In this case, the nodule gradually enlarged and we suspected a malignant tumor.
Bronchopulmonary endometriosis occurs during the child-bearing years and showed
various CT findings; nodules appeared in more elderly women compared with other
entities of thoracic endometrial syndrome. Joseph et al. supposed hormonal weakness
is not involved in aggressive findings such as cavity formation or surrounding
hemorrhage ( 1 ). This case
was a 49-year-old premenopausal woman. The pathological findings showed a soft
organization lacking constrictive fibrosis and with no marked menstrual hemorrhage
or tissue response.
Recently, the usefulness of MRI for the diagnosis of thoracic endometriosis was
reported ( 4 – 9 ). T1W imaging showed high signal intensity
and DWI was sensitive for small hematomas in pleurodiaphragmatic endometriosis
( 5 , 7 , 9 ). To the best of our knowledge, there has
been no report about MRI findings of bronchopulmonary endometriosis. In this case,
T1W and T2W imaging both showed high and low signal intensity and DWI showed
diffusion-restriction and enhancement areas in the nodule, probably caused by
hematoma during various periods. Low signal intensity in the part of the nodule
showed that contrast enhancement might correspond to endometrial tissue. Therefore,
these MRI findings are valuable for diagnosis. A signal void in the nodule may
reflect cavity formation or hemosiderin deposition. Pathological findings revealed
there was collection of red blood cell in dilated endometrial glands and a small
amount of hemosiderin or hemosiderin-laden macrophages were present in the
interstitium of the endometrial tissue. The nodule was composed of fresh hematoma
and hemosiderin deposition, so these findings might reflect various periods of
hemorrhage in MRI.
Pleurodiaphragmatic endometriosis and bronchopulmonary endometriosis also showed
hemorrhagic disease and showed similar signal intensities in MRI. The difference
between these two diseases can be seen where there is endometrial tissue in pleura
and the diaphragm or lung parenchyma. Bronchopulmonary endometriosis showed cavity
formation or GGO and consolidation in the form of pulmonary hemorrhage in CT.
However, catamenial hemoptysis is more important for diagnosis. MRI findings of this
case showed a hemorrhagic nodule with a partly enhanced area. These findings are not
specific in bronchopulmonary endometriosis and also show hematoma with granulation
tissue in pulmonary laceration, hemorrhagic nodules such as metastatic
choriocarcinoma, or angiosarcoma. If catamenial hemopysis is present, a diagnosis of
bronchopulmonary endometriosis is possible and past clinical histories are also
helpful for diagnosis.
We reported a case of bronchopulmonary endometriosis that occurred in the left lung.
The disease showed a gradually growing pulmonary nodule with an enhanced area. T1W
imaging, T2W imaging, and DWI showed signal intensity of hemorrhage during various
periods and enhancing areas associated with endometrial tissue.
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