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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">In 1977&#44; Engleman et al&#46; described pulmonary hyalinizing granuloma &#40;PHG&#41; as a separate entity&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> This disease is rare and is radiologically characterized by multiple and often bilateral nodules with no preferential localization&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Its etiology and pathogenesis remain unclear&#44; and a definitive diagnosis is provided by histopathological study of the lesions describing hyalinized lamellar collagen bundles surrounded by plasma cells&#44; lymphocytes&#44; and histiocytes&#46; This disease usually evolves with a benign course with nodules slowly increasing in size over years&#46; Specific treatment is not usually required&#44; although corticosteroids have demonstrated some efficacy&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The etiology of PHG is not well established&#44; and no relation to occupational exposure has been identified&#46; Due to the frequent association with infectious&#44; autoimmune&#44; and tumoral diseases&#44; an abnormal immune reaction has been proposed to explain the development of PHG&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#44;4</span></a> In the case in the present report&#44; we describe an uncommon association between PHG and a lymphoproliferative disorder&#46; We found only four similar reports of associations with multicentric Castleman disease<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a>&#59; diffuse lymphocytic lymphoma of the abdomen<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a>&#59; and pulmonary small lymphocytic lymphoma<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a>&#46; The ages of these patients ranged between 43 and 50 years&#44; and the patients were predominantly male &#40;3 males&#47;1 female&#41;&#46; All patients presented with multiple and bilateral pulmonary nodules&#44; and diagnosis was made through video-assisted thoracoscopic lung biopsy in three cases and by post-mortem examination in one case&#46; Chemotherapy schemes targeted at lymphoproliferative disease which included corticosteroid were initiated in three cases&#46; In two of these cases&#44; there was a reduction in the lung nodule dimensions&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">We report a case of a 69-year-old man&#44; former smoker &#40;70 pack-years&#41;&#44; with occupational exposure to dust and fumes from metallurgical casting and contact with birds&#44; who presented to the emergency department with a 6-month history of progressive dyspnea&#46; He reported mild fever&#44; anorexia&#44; and weight loss &#40;10&#8239;kg over the prior 3 months&#41;&#46; A physical examination revealed inspiratory crackles in the lower lungs&#46; Chest computed tomography &#40;CT&#41; scan showed bilateral peripheral consolidation areas&#44; mainly in the lower lobes but also involving the middle lobe and lingula &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a> &#8212; A and B&#41;&#59; the CT also revealed hepatosplenomegaly&#46; Laboratory data demonstrated normocytic and normochromic anemia&#44; and elevated levels of C-reactive protein and erythrocyte sedimentation rate&#46; An autoimmunity study was negative&#46; No endobronchial lesions were detected in the bronchoscopy&#46; In the bronchoalveolar lavage&#44; the total and differential cell counts revealed lymphocytic &#40;47&#37;&#41; and neutrophilic &#40;7&#46;4&#37;&#41; alveolitis&#44; which was negative for microorganisms and malignant cells&#46; An initial pulmonary function test revealed moderate restrictive ventilatory alteration with a forced expiratory volume in one second &#40;FEV1&#41; 2&#46;06&#8239;L &#40;72&#46;8&#37; of the predicted value&#41;&#44; forced vital capacity &#40;FVC&#41; 2&#46;42&#8239;L &#40;65&#46;6&#37;&#41;&#44; preserved FEV1&#47;FVC ratio&#44; a total lung capacity &#40;TLC&#41; 4&#46;51&#8239;L &#40;68&#46;5&#37;&#41;&#44; and carbon monoxide transfer factor &#40;TLCO&#41; 52&#37;&#46; A CT-guided transthoracic core biopsy was subsequently performed&#44; and hyalinized lamellar collagen tissue surrounded by lymphoplasmacytic infiltrates was reported on histological examination &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>E<span class="elsevierStyleSup">1</span>&#41;&#46; No evidence of neoplastic cells was found&#44; and acid-fast&#44; fungal&#44; and Congo red stains were all negative &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>E<span class="elsevierStyleSup">2</span>&#41;&#46; These findings were consistent with a diagnosis of PHG&#46; The patient was started on oral corticosteroid therapy &#40;deflazacort in an equivalent dose of 0&#46;5&#8239;mg&#47;kg&#47;day of prednisolone&#44; within a weaning scheme&#41; and demonstrated partial clinical and functional but not radiological improvement&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Throughout the investigation&#44; an indolent non-Hodgkin&#8217;s lymphoma &#40;NHL&#41; was incidentally diagnosed&#44; supported by a bone marrow biopsy&#46; The clinical case was&#44; therefore&#44; discussed in a multidisciplinary team&#44; and with PHG hypothesized as a paraneoplastic manifestation of hematological disease&#44; a decision to start NHL treatment was made&#46; After completing eight cycles of chemotherapy &#40;rituximab&#44; cyclophosphamide&#44; vincristine&#44; and prednisolone&#41;&#44; the patient had significant clinical&#44; functional&#44; and radiological improvement &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a> &#8212; B and D&#41;&#46; At the time this report was written&#44; the patient was asymptomatic and functional with FEV1 2&#46;88&#8239;L &#40;106&#46;5&#37;&#41;&#44; FVC 3&#46;66&#8239;L &#40;103&#46;1&#37;&#41;&#44; TLC 5&#46;83&#8239;L &#40;89&#46;7&#37;&#41;&#44; and TLCO 68&#37;&#46; No residual disease was found on further bone marrow tests&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In summary&#44; PHG is a rare entity with nonspecific symptoms and slow progression&#46; Although isolated or multiple nodular lesions are the most frequent findings&#44; PHG can occaisonally appear as lung parenchymal infiltration or consolidation with irregular or indistinct borders&#44; such as in this case&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In patients showing a rapid course and significant functional impairment&#44; corticosteroid treatment may be attempted&#44; despite the unclear efficacy of this approach&#46; Due to the frequent association with underlying diseases&#44; a careful investigation should be performed for therapeutic and prognostic implications&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical disclosures</span><p id="par0030" class="elsevierStylePara elsevierViewall">There are no personal details of patient in any part of the paper and in any supplementary materials &#40;including illustrations&#41;&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0035" class="elsevierStylePara elsevierViewall">No funding&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interests</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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Letter to the Editor
Pulmonary hyalinizing granuloma: Atypical presentation
Â.D. Cunhaa,
Corresponding author
6979@hstviseu.min-saude.pt

Corresponding author.
, N. Melob, S. Guimarãesc, C.S. Mourac, J.M. Pereirad, A. Moraisb,e,f
a Pulmonology Department, Centro Hospitalar Tondela-Viseu, Viseu, Portugal
b Pulmonology Department, Centro Hospitalar São João, Porto, Portugal
c Pathology Department, Centro Hospitalar São João, Porto, Portugal
d Radiology Department, Centro Hospitalar São João, Porto, Portugal
e Faculdade de Medicina do Porto, University of Porto, Portugal
f Institute for Research and Innovation in Health (I3S), University of Porto, Portugal
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and no relation to occupational exposure has been identified&#46; Due to the frequent association with infectious&#44; autoimmune&#44; and tumoral diseases&#44; an abnormal immune reaction has been proposed to explain the development of PHG&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#44;4</span></a> In the case in the present report&#44; we describe an uncommon association between PHG and a lymphoproliferative disorder&#46; We found only four similar reports of associations with multicentric Castleman disease<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a>&#59; diffuse lymphocytic lymphoma of the abdomen<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a>&#59; and pulmonary small lymphocytic lymphoma<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a>&#46; The ages of these patients ranged between 43 and 50 years&#44; and the patients were predominantly male &#40;3 males&#47;1 female&#41;&#46; All patients presented with multiple and bilateral pulmonary nodules&#44; and diagnosis was made through video-assisted thoracoscopic lung biopsy in three cases and by post-mortem examination in one case&#46; Chemotherapy schemes targeted at lymphoproliferative disease which included corticosteroid were initiated in three cases&#46; In two of these cases&#44; there was a reduction in the lung nodule dimensions&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">We report a case of a 69-year-old man&#44; former smoker &#40;70 pack-years&#41;&#44; with occupational exposure to dust and fumes from metallurgical casting and contact with birds&#44; who presented to the emergency department with a 6-month history of progressive dyspnea&#46; He reported mild fever&#44; anorexia&#44; and weight loss &#40;10&#8239;kg over the prior 3 months&#41;&#46; A physical examination revealed inspiratory crackles in the lower lungs&#46; Chest computed tomography &#40;CT&#41; scan showed bilateral peripheral consolidation areas&#44; mainly in the lower lobes but also involving the middle lobe and lingula &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a> &#8212; A and B&#41;&#59; the CT also revealed hepatosplenomegaly&#46; Laboratory data demonstrated normocytic and normochromic anemia&#44; and elevated levels of C-reactive protein and erythrocyte sedimentation rate&#46; An autoimmunity study was negative&#46; No endobronchial lesions were detected in the bronchoscopy&#46; In the bronchoalveolar lavage&#44; the total and differential cell counts revealed lymphocytic &#40;47&#37;&#41; and neutrophilic &#40;7&#46;4&#37;&#41; alveolitis&#44; which was negative for microorganisms and malignant cells&#46; An initial pulmonary function test revealed moderate restrictive ventilatory alteration with a forced expiratory volume in one second &#40;FEV1&#41; 2&#46;06&#8239;L &#40;72&#46;8&#37; of the predicted value&#41;&#44; forced vital capacity &#40;FVC&#41; 2&#46;42&#8239;L &#40;65&#46;6&#37;&#41;&#44; preserved FEV1&#47;FVC ratio&#44; a total lung capacity &#40;TLC&#41; 4&#46;51&#8239;L &#40;68&#46;5&#37;&#41;&#44; and carbon monoxide transfer factor &#40;TLCO&#41; 52&#37;&#46; A CT-guided transthoracic core biopsy was subsequently performed&#44; and hyalinized lamellar collagen tissue surrounded by lymphoplasmacytic infiltrates was reported on histological examination &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>E<span class="elsevierStyleSup">1</span>&#41;&#46; No evidence of neoplastic cells was found&#44; and acid-fast&#44; fungal&#44; and Congo red stains were all negative &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>E<span class="elsevierStyleSup">2</span>&#41;&#46; These findings were consistent with a diagnosis of PHG&#46; The patient was started on oral corticosteroid therapy &#40;deflazacort in an equivalent dose of 0&#46;5&#8239;mg&#47;kg&#47;day of prednisolone&#44; within a weaning scheme&#41; and demonstrated partial clinical and functional but not radiological improvement&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Throughout the investigation&#44; an indolent non-Hodgkin&#8217;s lymphoma &#40;NHL&#41; was incidentally diagnosed&#44; supported by a bone marrow biopsy&#46; The clinical case was&#44; therefore&#44; discussed in a multidisciplinary team&#44; and with PHG hypothesized as a paraneoplastic manifestation of hematological disease&#44; a decision to start NHL treatment was made&#46; After completing eight cycles of chemotherapy &#40;rituximab&#44; cyclophosphamide&#44; vincristine&#44; and prednisolone&#41;&#44; the patient had significant clinical&#44; functional&#44; and radiological improvement &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a> &#8212; B and D&#41;&#46; At the time this report was written&#44; the patient was asymptomatic and functional with FEV1 2&#46;88&#8239;L &#40;106&#46;5&#37;&#41;&#44; FVC 3&#46;66&#8239;L &#40;103&#46;1&#37;&#41;&#44; TLC 5&#46;83&#8239;L &#40;89&#46;7&#37;&#41;&#44; and TLCO 68&#37;&#46; No residual disease was found on further bone marrow tests&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In summary&#44; PHG is a rare entity with nonspecific symptoms and slow progression&#46; Although isolated or multiple nodular lesions are the most frequent findings&#44; PHG can occaisonally appear as lung parenchymal infiltration or consolidation with irregular or indistinct borders&#44; such as in this case&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In patients showing a rapid course and significant functional impairment&#44; corticosteroid treatment may be attempted&#44; despite the unclear efficacy of this approach&#46; Due to the frequent association with underlying diseases&#44; a careful investigation should be performed for therapeutic and prognostic implications&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical disclosures</span><p id="par0030" class="elsevierStylePara elsevierViewall">There are no personal details of patient in any part of the paper and in any supplementary materials &#40;including illustrations&#41;&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0035" class="elsevierStylePara elsevierViewall">No funding&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interests</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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Article information
ISSN: 25310437
Original language: English
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Pulmonology

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