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A case of pulmonary eosinophilic granuloma that remitted after
cessation of smoking.Nihon
Kokyuki Gakkai Zasshi. 2006
Jul;44(7):528-31.
A 43-year-old man
was admitted to our hospital because of productive cough and an
abnormal chest radiograph. He had smoked one pack of cigarettes per
day for 23 years. Chest radiograph on admission showed diffuse
reticulo-linear infiltrates, most prominent in both upper lung fields.
Chest HRCT scan showed multiple micronodules, cavitated nodules, and
thick-walled cysts with ground-glass attenuation. These radiographic
findings suggested pulmonary eosinophilic granuloma (PEG).
Histological findings of the biopsy specimen obtained from
video-assisted thoracoscopy demonstrated granulomatous lesions
consisting of S-100 protein-positive Langerhans cells with
infiltration of eosinophils and fibrous lesions (starfish-like
fibrosis). A definitive diagnosis of PEG was made, and he immediately
ceased smoking. The symptoms and radiographic findings markedly
improved within 3 months after cessation of smoking. Chest HRCT on the
final scan revealed that nodules and almost all cystic lesions
vanished. He resumed smoking without any sign of recurrence.
Obstructing
tracheal pulmonary Langerhans cell histiocytosis.Chest.
2005 Aug;128(2):1057-8.
Pulmonary
Langerhans cell histiocytosis, also known as eosinophilic granuloma,
is an uncommon interstitial lung disease. A solitary nodule, usually
parenchymal, may rarely be the only manifestation of the disease. We
describe a case of Langerhans cell histiocytosis presenting as an
obstructing tracheal lesion in a 55-year-old woman. Following complete
resection of the lesion via flexible bronchoscopy, full recovery was
achieved. This case represents a unique cause for tracheal
obstruction, as well as an unreported manifestation of pulmonary
Langerhans cell histiocytosis.
Pulmonary
eosinophilic granuloma presenting as a solitary pulmonary nodule.Nihon
Kokyuki Gakkai Zasshi. 2005
Jan;43(1):37-40.
A symptomless
60 year old man with a heavy smoking history presented with a nodule
in the right lung, which was found by an annual chest X-ray. Chest CT
showed a 1 cm speculated nodule in the right S8. Bronchoscopic
transbronchial biopsy to the nodule yielded no definite diagnosis.
Since the nodule was highly suggestive of lung cancer, wedge resection
was performed by video-assisted thoracoscopic surgery. Pathological
findings of the specimen showed that the nodule consisted of a mixed
population of histiocytoid cells with eosinophils. The nuclei and
cytoplasms of the histiocytoid cells were stained positively for S-100
protein. Pulmonary eosinophilic granuloma (PEG) was diagnosed. Common
radiographic findings of PEG should present with a mixture of multiple
nodular shadows and cystic lesions. PEG presenting as a solitary
nodule is rare.
Two
cases of pulmonary eosinophilic granuloma with multiple nodular
shadows.Nihon
Kokyuki Gakkai Zasshi. 2002
Dec;40(12):984-8.
We encountered
two rare cases of pulmonary eosinophilic granuloma with multiple
nodular shadows in both lungs. The patient in case 1 was a 54-year-old
man complaining of dry cough and chest pain. He had smoked 20
cigarettes a day for 36 years. The patient in case 2 was a 37-year-old
woman complaining of dry cough. She had smoked 15 cigarettes a day for
20 years. Chest radiography and CT revealed multiple nodular shadows
in both lungs. Diagnosis was made by open lung biopsy in case 1 and by
percutaneous lung biopsy in case 2. After smoking cessation, symptoms
improved markedly and the shadows in the chest radiographs and CT
disappeared. In cases of bilateral multiple nodular shadows, other
than metastatic lung tumor cases, pulmonary eosinophilic granuloma
should be considered.
Clinical
course of pulmonary eosinophilic granuloma--CT evidence of lesion
evolution.Nihon
Kokyuki Gakkai Zasshi. 2002
Nov;40(11):856-62.
We reviewed a
clinicopathological study of pulmonary eosinophilic granuloma focusing
on the evolution of pulmonary lesions in chest CT scans. Between 1990
and 2002, five patients with pulmonary eosinophilic granuloma were
admitted to our hospital. Clinical features, chest radiographs,
pathological characteristics and treatments were evaluated. The scans
were repeated in four patients at intervals of at least eight months.
All patients were men who smoked. The age at onset of the pulmonary
disorders ranged from 22 to 45 years, with a mean of 33 years. Most of
the initial chest CT scans showed nodular lesions and thin-walled
cysts, but the nodular lesions disappeared or were transformed into
cystic lesions in the later scans. Most of the final scans revealed
thin-walled cysts and emphysematous lesions. The histopathological
characteristics of the nodular lesions in the chest CT were correlated
with cellular granulomas mainly composed of Langerhans cells and
eosinophils, and small granulomas were also seen in the thin fibrous
walls of the cysts. All patients quitted smoking and three showed
improvement of the pulmonary lesions within three months.
Improvement of extrapulmonary lesions of eosinophilic granuloma
after cessation of smoking.Nihon
Kokyuki Gakkai Zasshi. 2002
Apr;40(4):326-30
We encountered
a case of pulmonary eosinophilic granuloma complicated with
pneumothorax. A 24-year-old man was admitted to our department because
of respiratory difficulty. Chest radiography on admission showed a
right pneumothorax. A thoracic catheter was therefore inserted, and
the pneumothorax was improved. A chest radiograph obtained after
treatment showed diffuse linear reticular shadows; and a chest CT scan
showed starlike nodular lesions and multiple cysts in the lungs, and a
large nodule in the left rib. Because an ulcer had been diagnosed in
the oral cavity 1 year previously, a mandible biopsy was performed,
and a granulomatous lesion consisting of eosinophils was recognized.
Taken together with the pulmonary findings, an eosinophilic granuloma
was diagnosed. The patient was instructed not to smoke and the
clinical course was observed. The pulmonary, mandibular, and costal
lesions improved.
Pulmonary
eosinophilic granuloma in a woman of 72.Nihon
Kokyuki Gakkai Zasshi. 2002
Apr;40(4):316-20.
We report a
case of pulmonary eosinophilic granuloma with the latest onset ever
reported in Japan. A 72-year-old woman, a current smoker, developed
dyspnea and dry cough in late January 2001. Chest radiography and CT
examinations revealed numerous small nodular lesions and multiple
cysts in both upper lung fields. A thoracoscopic lung biopsy revealed
a granulomatous lesion composed of eosinophils and large histiocytic
cells with pale eosinophilic cytoplasms and large cleaved nuclei.
These cells gave a positive reaction for S-100 protein. These findings
led to a diagnosis of pulmonary eosinophilic granuloma. This disease
is generally reported in much younger subjects, and few cases are
known in those over 60-years old. No Japanese report of histologically
confirmed pulmonary eosinophilic granuloma in an older patient has
previously appeared, but it may now be prudent to take this disease
into consideration when diagnosing pulmonary disease in older
subjects.
A case of
pulmonary eosinophilic granuloma arising rapidly 30 years after the
start of smoking and remitting spontaneously without smoking
cessation.Nihon
Kokyuki Gakkai Zasshi. 2001
Nov;39(11):852-6.
A case of
pulmonary eosinophilic granuloma which arose rapidly after 30 years of
smoking and remitted spontaneously without smoking cessation is
reported. The patient was a 54-year-old man complaining of a dry cough
who had been smoking 30 cigarettes a day for 30 years. Chest
roentgenography showed multiple nodular shadows and cystic lesions in
the upper and middle fields of both lungs. Chest computed tomography
revealed multiple small cysts and small nodular lesions, mainly in
both upper lung fields. CT findings strongly suggested pulmonary
eosinophilic granuloma. A transbronchial lung biopsy (TBLB) was
performed and 4 specimens were obtained, of which 3 showed
granulomatous lesions with eosinophils and histiocytes. Furthermore,
the granulomatous lesions were positive for S-100 protein staining.
The symptoms and radiographic findings improved markedly within about
6 months after the onset of symptoms without treatment. Many cases of
this disease were diagnosed in the past by open lung biopsy, but the
number of cases diagnosed by TBLB is now increasing. The effectiveness
of open lung biopsy has been emphasized in the diagnosis of pulmonary
eosinophilic granuloma, but TBLB is also useful for diagnosis,
especially in the active or early stage of the disease.
Three cases of
pulmonary eosinophilic granuloma.Nihon
Kokyuki Gakkai Zasshi. 2001
May;39(5):333-7
CASE 1. A
55-year-old man was admitted because of an abnormality in chest
radiographs. Chest HRCT showed multiple cystic lesions with thick and
thin walls, and nodules; and strongly suggested pulmonary eosinophilic
granuloma (EG). Open lung biopsy revealed granuloma formation and a
fibrotic area consistent with EG. CASE 2. A 28-year-old woman was
admitted because of chest pain and dyspnea. Chest radiography revealed
bilateral pneumothorax. Chest HRCT showed multiple cystic lesions with
thick walls involving the entire lung. As lung biopsy revealed,
proliferative lesions and cavitation containing S-100 protein-positive
histiocytes, we diagnosed this case as pulmonary EG in the active
stage. CASE 3. A 32-year-old woman was admitted because of dyspnea.
Chest CT showed bullous changes in the lung. As an open lung biopsy
revealed, honeycomb changes with S-100 protein-positive histiocytes,
this case was diagnosed as the regressive phase of EG. It is known
that the chest radiography and HRCT findings of EG are characteristic
and vary with the stage, so a surgical lung biopsy is necessary for
diagnosis of EG. It is considered that the prognosis of the active
phase of EG involving the entire lung is poor.
Pneumothorax
secondary to pulmonary histiocytosis X.Minerva
Chir. 1999 Jul-Aug;54(7-8):531-6.
The authors
report a case of pneumothorax in a young man suffering from pulmonary
histiocytosis. They take this observation as a starting point for
discussing about the so-called "histiocytosis X". The term "histiocytosis"
is a term for a variety of proliferative disorders of histiocytes or
macrophages. There is a small cluster of conditions characterized by
proliferation of a special type of histiocyte called the "Langerhans'
cell". The proliferation of Langerhans' cells results from
disturbances in immunoregulation. Particularly, there are three
clinicopathologic entities: *Letterer-Siwe disease (acute disseminated
Langerhans' cells histiocytosis), *multifocal eosinophilic granuloma
or Hand-Schüller-Christian disease (multifocal Langerhans' cells
histiocytosis), *unifocal eosinophilic granuloma (unifocal Langerhans'
cells histiocytosis). In the past, these were collected in the not
specific term of histiocytosis X. Most patients with Langerhans' cell
granulomatosis have diseases limited to the lung, only a small
proportion have extrapulmonary involvement or a systemic disorder.
Typically, there are expanding, erosive accumulations of Langerhans'
cells within the medullary cavities of bones. In this reported case, a
patient, two years before, was affected by osteolytic bone lesion in
the left femur. Pulmonary histiocytosis is often seen in smokers in
the third and fourth decade of life. The 20% of patients are
asymptomatic and found to have an abnormal chest radiograph; a lesser
proportion presents with pneumothorax. Some patients develop
progressive fibrotic lung disease and respiratory failure. Chest
radiographs may show bilateral reticular or reticulonodular
infiltrates, sometimes with cystic changes, often with sparing of the
costophrenic angles, but high resolution computerized tomography scans
may be virtually diagnostic based on the predilection of the cysts and
nodules for the upper lung zones. Sometimes the typical Langerhans'
cells may be recovered in bronchoalveolar lavage fluid of patients
with active disease. The combination of the characteristic CT findings
and Langerhans' cells in bronchoalveolar lavage fluid should be
diagnostic, decreasing the number of cases requiring for diagnosis
open lung biopsy. The differential diagnosis is with sarcoidosis, tbc,
diffuse metastases, lymphoma, eosinophilic pneumonia, fibrosing
interstitial pneumonia.
Pulmonary
eosinophilic granuloma--clinical analysis of 17 patients.Nihon
Kyobu Shikkan Gakkai Zasshi. 1995
Dec;33(12):1372-1381.
We studied the
clinical features of 17 Japanese patients with pulmonary eosinophilic
granuloma. Fourteen of the patients were men and three were women;
they ranged in age from 19 to 64 years, with a mean of 34 years at the
time of the first examination. Pathologic diagnosis in all patients
was based on histologic findings of specimens obtained by open lung
biopsy. Major symptoms were dry or productive cough, chest pain,
dyspnea, and fever; 23.3% of the patients were asymptomatic. Five
patients had pneumothorax. Most patients did not have abnormal
physical signs. All 17 patients had histories of smoking, and 14 had
started to smoke cigaretts before the age of 20 years. Ten patients
(58.8%) first presented with cough or dyspnea, and in the other
patients (41.2%) the first abnormalities detected were pulmonary
infiltrates on chest radiographs during health examinations. Chest
roentgenograms usually showed bilateral abnormalities. These
abnormalities were distributed over all lung fields in 9 cases
(52.9%), in the upper and middle lung fields in 4 cases (23.5%) in the
upper lung fields in 3 cases (17.7%), and in the middle lung fields in
1 case (5.9%). Micronodular, reticular, cystic or linear shadows were
evident in most cases, and were mixed in various proportions. Eleven
patients (65%) had abnormalities of pulmonary function. Low %VC and
%FEV1 and high RV/TLC ratios were observed in 20-40% of the patients.
Low DLCOs (%DLCO < 70%) were observed in 53% of the patients. Arterial
blood gases were normal in 11 of 15 patients. The extent of shadows in
the chest roentgenogram was related to the frequency of dyspnea, to
the total number of cells in bronchoalveolar lavage fluid, and to the
abnormally low %FVC and %FEV1, but not to the %DLCO. Data from
bronchoalveolor lavage fluid were non-specific in this disease, but
further studies will be needed. Follow-up data were collected on 16
patients. The mean time from the histologic diagnosis after open lung
biopsy to the last observation was 81.8 +/- 45.1 months (range, 2
months to 15 years). One patient died of pulmonary eosinophilic
granuloma. The usefulness of steroid therapy remains uncertain.
A case of
pulmonary eosinophilic granuloma and diabetes insipidus.Nihon
Kyobu Shikkan Gakkai Zasshi. 1995
May;33(5):576-82.
A 31-year-old
man was admitted to our hospital because of a sudden onset of thirst,
polyposia, and polyuria. Five years previously he had been admitted to
our hospital because of a dry cough. On the first admission, the chest
X-ray film had shown reticular shadows and bullous changes in both
upper lung fields. Histological examination of a transbronchial lung
biopsy specimen had revealed that the nodular lesion in the
interstitium of the alveolar lesion consisted of an aggregate of many
Langerhans cells with pale cytoplasm and partly convoluted nuclei. In
addition, immunoperoxidase stain for S-100 protein had been strongly
positive in numerous Langerhans cells in a bone biopsy specimen from a
left mandibullar lesion, which is the same histological appearance as
the lung lesion. A diagnosis of pulmonary eosinophilic granuloma had
been made. The course after discharge was not progressive without
treatment for 5 years, but the patient suddenly began to have thirst,
polyposia, and polyuria. Dehydration, vasopressin tests, and the
findings of MRI indicated diabetes insipidus due to a pathological
change in the pituitary gland. Although diabetes insipidus is known to
be a common complication of pulmonary eosinophilic granuloma, only 9
cases have been reported in Japan.
Transbronchial
biopsy in patients with pulmonary eosinophilic granuloma. Comparison
with findings on open lung biopsy.Arch
Pathol Lab Med. 1994 May;118(5):523-30.
We evaluated
the findings on transbronchial biopsy specimens in reference to open
lung biopsy specimens from 12 patients with pulmonary eosinophilic
granuloma. Features in transbronchial biopsy specimens were further
contrasted to those of patients with interstitial fibrosis and
nondiagnostic biopsy specimens for localized lesions. Transbronchial
biopsy specimens were randomized and graded for histologic features
and cellularity. Patients with eosinophilic granuloma had more
macrophages (P < .05) but equivalent numbers of eosinophils,
neutrophils, and Langerhans' cells compared with those of the other
two groups. Only two endoscopic biopsy specimens were histologically
diagnostic or highly consistent with eosinophilic granuloma. We
conclude that the diagnosis of eosinophilic granuloma is possible on
transbronchial biopsy but requires a high index of suspicion. The
demonstration of Langerhans' cells by immunohistochemical staining for
S100 protein is a useful adjunct. The low diagnostic yield for
eosinophilic granuloma on transbronchial biopsy results from
inadequate sampling and from the nonspecific appearance of discrete
lesions in small tissue samples.
A case of
pulmonary eosinophilic granuloma confirmed by transbronchial lung
biopsy.
Nihon
Kyobu Shikkan Gakkai Zasshi. 1994
Jan;32(1):111-5.
A case of
pulmonary eosinophilic granuloma confirmed by transbronchial lung
biopsy (TBLB) is reported. A 63-year-old man was admitted to our
hospital complaining of productive cough and exertional dyspnea. Chest
roentgenogram and computed tomography showed bilateral linear and
reticular shadows predominantly in the upper and middle lung fields.
TBLB was performed and the specimens showed granulomatous lesions
consisting of histiocytes with infiltration of eosinophils.
Histiocytes were positive for S-100 protein by immunohistochemistry.
He was diagnosed as having pulmonary eosinophilic granuloma. In the
diagnosis of pulmonary eosinophilic granuloma, open lung biopsy is
useful, but TBLB is also useful for diagnosis in the active or early
stage of this disease.
Immunohistochemical and ultrastructural examination of histiocytosis X
cells in pulmonary eosinophilic granuloma.Acta
Pathol Jpn. 1992 Oct;42(10):719-26.
We describe
histological, immunohistochemical, and ultrastructural findings in
pulmonary eosinophilic granuloma (PEG) from three patients. The
specimens were taken by open-lung biopsy. The lesions of the lung were
composed of histiocytic cells, macrophages and eosinophils. The
histiocytic cells reacted positively with anti-S-100 protein antibody.
The histiocytic cells had various types of Birbeck granules in the
cytoplasm. The histiocytic cells were histiocytosis X (HX) cells
considered to be derived from Langerhans cells. Sporadic mitosis of HX
cells was observed. Some HX cells had migrated from the lesions into
the alveolar spaces through the alveolar cell layers. In the lesions
of PEG, HX cells have self-reproduction and migration capability.
Neoplasms
associated with pulmonary eosinophilic granuloma.Arch
Pathol Lab Med. 1991 May;115(5):499-506.
We found
a high prevalence of pulmonary and extrapulmonary neoplasms in
patients with pulmonary eosinophilic granuloma (PEG) who were studied
at our institution. Among 21 patients with PEG, 10 (48%) had
associated benign (one patient) or malignant (nine patients) tumors.
Patients with tumors were older at the time of diagnosis of PEG (48.9
vs 34.5 years). Tumors included three lung carcinomas, one pulmonary
carcinoid tumor, two lymphomas, five extrapulmonary carcinomas, and
one mediastinal ganglioneuroma. Two malignant neoplasms developed in
each of two patients. Six tumors preceded, three followed, and three
occurred concomitantly with the diagnosis of PEG. Slides from eight
PEG-associated tumors and 18 control neoplasms from patients without
PEG were also stained immunohistochemically for S100 protein. Four
PEG-associated (50%) and 11 control (61%) tumors contained S100
protein-positive interstitial cells. Our study suggests, but does not
prove, that there may be more than a random association between PEG
and neoplasms. Cigarette smoking, moreover, is an important risk
factor for both PEG and lung carcinomas. Our immunohistochemical
findings indicate that S100 protein-positive cells in tumors usually
bear little or no relationship to PEG. In patients with an underlying
malignant neoplasm, PEG simulates pulmonary metastases clinically and,
occasionally, histopathologically.
Increased pulmonary neuroendocrine cells with bombesin-like
immunoreactivity in adult patients with eosinophilic granuloma.J
Clin Invest. 1990 Sep;86(3):838-44.
Cigarette
smoking is associated with hyperplasia of pulmonary neuroendocrine
cells and variably increased levels of bombesin-like peptides in the
lower respiratory tract. Because the neuropeptide bombesin is a
chemoattractant for monocytes and a mitogen for 3T3 fibroblasts, we
hypothesized that an excess of neuroendocrine cells and bombesin-like
peptides could contribute to lung inflammation and fibrosis in certain
cigarette smokers. Eosinophilic granuloma is a fibrotic lung disease
of unknown etiology that in adults occurs almost invariably in
cigarette smokers. We quantitated neuroendocrine cells with bombesin-like
immunoreactivity in open lung biopsies from patients with eosinophilic
granuloma (n = 6) and compared these with cigarette smokers (n = 6)
who underwent lung resection for reasons other than primary lung
disease. In addition, we compared them with patients with idiopathic
pulmonary fibrosis (n = 8), a disease not associated with cigarette
smoking. Finally, we also examined the mitogenic effect of bombesin on
cultured human adult lung fibroblasts. The patients with eosinophilic
granuloma exhibited a 10-fold increase in neuroendocrine cells with
bombesin-like immunoreactivity compared to both smokers (P = 0.005)
and patients with idiopathic pulmonary fibrosis (P = 0.005). In
addition, bombesin produced a significant mitogenic effect on cultured
human adult lung fibroblasts at concentrations of 1 nM and above. We
conclude that increased numbers of pulmonary neuroendocrine cells with
bombesin-like immunoreactivity are commonly found in patients with
eosinophilic granuloma and, since bombesin-like peptides are
chemotactic for monocytes and mitogenic for human lung fibroblasts, we
speculate that neuroendocrine cell hyperplasia may be important in the
pathogenesis of eosinophilic granuloma in adult cigarette smokers.
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