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    "textoCompleto" => "<p class="elsevierStylePara"><a name="sec0005" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Introduction</span><p class="elsevierStylePara"> Idiopathic pleuroparenchymal fibroelastosis &#40;IPPFE&#41; is a rare condition characterized by fibroelastotic thickening of the pleural and subpleural lung parenchyma&#44; mainly in the upper lobes&#46;<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a> This entity was first mentioned in English-language medical literature by Frankel et al&#46;<a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> in 2004&#44; although the same concept designated as idiopathic pulmonary upper lobe fibrosis was proposed in 1992 by Aminati et al&#46;<a href="&#35;bib3" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> There is little known about its etiology and most cases are considered to be idiopathic&#44; although a few cases are believed to occur in a familial&#47;genetic lung disease context<a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> and others have been reported in association with previous bone marrow transplantation&#46;<a href="&#35;bib4" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">4</span></a> New data have been published suggesting that recurrent infections may have a role in its pathogenesis&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> Recently IPPFE was recognized as a specific rare idiopathic interstitial pneumonia &#40;IIP&#41; in the update of the international multidisciplinary classification of the IIPs<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a>&#59; IPPFE was characterized by an elastotic fibrosis present with an intra-alveolar fibrosis&#46;<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a> High-resolution chest tomography &#40;HRCT&#41; shows dense subpleural consolidation with traction bronchiectasis&#44; architectural distortion&#44; and upper lobe volume loss&#46;<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a> The authors present two cases of IPPFE in which the final diagnosis was obtained after CT-guided transthoracic core lung biopsy&#46;</p><p class="elsevierStylePara"><a name="sec0010" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Case report 1</span><p class="elsevierStylePara"> The patient was a 66-year-old female&#44; caucasian&#44; nonsmoker&#44; had worked as art gallery assistant&#44; had a history of esophageal hiatus hernia and was chronically medicated with omeprazole&#46; She had no known family history of pulmonary disease&#46; In September 2010&#44; she complained of dyspnea on exertion and nonproductive cough&#44; with progressive worsening&#46; The patient was examined by a general practitioner and a chest X-ray was performed that revealed a reticular densification with peripheral distribution and predominance in the upper lobes&#46; Lung function tests were as follows&#58; FVC 1&#46;74&#160;L &#40;79&#46;4&#37; predicted&#41;&#44; FEV1 1&#46;51&#160;L &#40;83&#46;5&#37; predicted&#41;&#44; FEV1&#47;FVC 86&#46;8&#37;&#44; TLC 4&#46;16&#160;L &#40;95&#46;2&#37; predicted&#41;&#44; DLco 2&#46;85&#160;mL&#47;min&#47;mmHg &#40;44&#46;6&#37; predicted&#41;&#46; Blood gas parameters were in the reference range values&#46; She walked 450&#160;m at 6-min walk test &#40;6-WT&#41; with an oxygen desaturation of 12&#37; &#40;initial SatO2-97&#37; and final SatO2-85&#37;&#41;&#46; HRCT images showed pleural and subpleural thickening with fibrotic changes in the marginal parenchyma&#44; mainly in the upper lobes &#40;<a href="&#35;f0005" class="elsevierStyleCrossRefs">Figure 1</a>&#41;&#46; A short course of corticosteroids was prescribed with no clinical improvement&#46; The patient was then referred to the Interstitial Lung Disease outpatient clinic&#46; On chest examination&#44; fine bibasilar inspiratory rales were identified&#46; Autoimmune serological testing was negative&#46; A bronchoscopy was performed with no noticeable airways abnormalities&#46; Bronchoalveolar lavage revealed a lymphocytosis &#40;28&#46;4&#37;&#41; with a very high CD4&#47;CD8 ratio &#40;14&#46;5&#41;&#44; and no microorganism or malignant cells were identified&#46; Transbronchial and bronchial biopsies had no evidence of malignancy or granulomas&#46; The patient underwent a CT-guided transthoracic core lung biopsy in two distinct locations&#44; complicated by pneumothorax with bronchopleural fistula and subcutaneous emphysema&#46; Histological evaluation showed marked thickened visceral pleura and prominent predominantly elastic sub-pleural fibrosis with mild&#44; patchy lymphoplasmacytic inflammatory infiltrate&#59; orcein stain showed that the elastosis was within the alveolar walls&#44; marking the intra-alveolar fibrosis &#40;<a href="&#35;f0010" class="elsevierStyleCrossRefs">Figure 2</a>&#41;&#46; Despite the prescription of azathioprine &#40;2&#160;mg&#47;kg&#47;day&#41;&#44; the patient is clinically and functionally worse after 20 months of follow-up&#58; FVC 1&#46;63&#160;L &#40;82&#37; predicted&#41;&#44; FEV1 1&#46;44&#160;L &#40;89&#37; predicted&#41;&#44; FEV1&#47;FVC 89&#46;5&#37;&#44; TLC 3&#46;28&#160;L &#40;80&#37; predicted&#41;&#44; DLco 1&#46;66&#160;mL&#47;min&#47;mmHg &#40;26&#37; predicted&#41;&#46;</p><p class="elsevierStylePara"><a name="f0005" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img alt="High-resolution chest tomography in patient no&#46; 1 shows pleural and subpleural thickening with fibrotic changes in the marginal parenchyma&#44; mainly in the upper lobes&#46;" src="420v21n01-90385615fig1.jpg"></img></p><p class="elsevierStylePara"> Figure 1&#46; High-resolution chest tomography in patient no&#46; 1 shows pleural and subpleural thickening with fibrotic changes in the marginal parenchyma&#44; mainly in the upper lobes&#46;</p><p class="elsevierStylePara"><a name="f0010" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img src="420v21n01-90385615fig2.jpg"></img></p><p class="elsevierStylePara"> Figure 2&#46; Histological features of patient no&#46; 1 shows a sub-pleural elastotic fibrosis highlighted by the orcein elastic stain&#44; that marks the alveolar walls elastosis &#40;&#916;&#41; and the intra-alveolar fibrosis &#40;<a name="0" class="elsevierStyleCrossRefs"></a><img src="420v21n01-90385615figfx1.jpg"></img> &#41; &#40;H&#38;E&#44; 100&#215;&#59; Orcein&#44; 40&#215;&#41;&#46;</p><p class="elsevierStylePara"><a name="sec0015" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Case report 2</span><p class="elsevierStylePara"> The patient was a 59-year-old female&#44; caucasian&#44; nonsmoker&#44; accountant&#44; with history of pulmonary tuberculosis &#40;5-years-old&#41;&#44; post-infectious bronchiectasis&#44; recurrent respiratory infections&#44; and osteoporosis&#46; The patient had no known family history of pulmonary disease&#46; Since the age of 50 she had attended a respiratory outpatient clinic because of dyspnea on exertion and pleural thickening in the upper lobes&#46; These findings were interpreted as within the clinical picture of sequelae of tuberculosis and the patient was medicated with an inhaled association of budesonide and formoterol&#46; However&#44; after 7 years of follow-up&#44; the patient presented a worsening of dyspnea on exertion and dry cough&#46; On chest examination&#44; fine bibasilar inspiratory rales were identified&#46; Chest X-ray revealed a linear densification with peripheral distribution and predominance in the upper lobes&#44; with significant worsening compared to previous ones&#46; HRCT showed a pleural thickening with associated subpleural fibrosis&#44; mainly in the upper lobes &#40;<a href="&#35;f0015" class="elsevierStyleCrossRefs">Figure 3</a>&#41;&#46; Autoimmune serological testing was negative&#46; Lung function tests showed moderate restrictive ventilatory impairment and a significant decrease in diffusion capacity&#58; FVC 1&#46;37&#160;L &#40;56&#46;1&#37; predicted&#41;&#44; FEV1 1&#46;17&#160;L &#40;57&#46;3&#37; predicted&#41;&#44; FEV1&#47;FVC 85&#46;5&#37;&#44; TLC 2&#46;87&#160;L &#40;64&#46;7&#37; predicted&#41;&#44; DLco 2&#46;07&#160;mL&#47;min&#47;mmHg &#40;29&#46;4&#37; predicted&#41;&#46; Blood gas parameters were in the reference range values&#46; She walked 435&#160;m at 6-WT with an oxygen desaturation of 10&#37; &#40;initial SatO2-96&#37; and final SatO2-86&#37;&#41;&#46; A bronchoscopy was performed with no noticeable airways abnormalities&#46; Bronchoalveolar lavage revealed neutrophilic &#40;53&#46;8&#37;&#41; and eosinophilic &#40;14&#37;&#41; alveolitis and no microorganism or malignant cells were identified&#46; CT-guided transthoracic core lung biopsy was then performed with iatrogenic pneumothorax&#46; Histological evaluation showed marked thickened visceral pleura and sharply demarcated elastic fibrosis&#44; without significant inflammation&#59; orcein stain showed elastosis of the alveolar walls with a predominant intra-alveolar fibrosis &#40;<a href="&#35;f0020" class="elsevierStyleCrossRefs">Figure 4</a>&#41;&#46; Treatment was started with 400&#160;mg&#47;day of hydroxychloroquine and 7&#46;5&#160;mg&#47;day of prednisolone&#46; At the time of writing&#44; after a 10-month follow-up period&#44; the patient is clinically and functionally stable&#58; FVC 1&#46;27&#160;L &#40;54&#37; predicted&#41;&#44; FEV1 1&#46;07&#160;L &#40;54&#37; predicted&#41;&#44; FEV1&#47;FVC 84&#46;4&#37;&#44; TLC 2&#46;94&#160;L &#40;68&#37; predicted&#41;&#44; DLco 1&#46;52&#160;mL&#47;min&#47;mmHg &#40;22&#37; predicted&#41;&#46;</p><p class="elsevierStylePara"><a name="f0015" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img alt="High-resolution chest tomography in patient no&#46; 2 shows a pleural thickening with associated subpleural fibrosis&#44; mainly in the upper lobes&#46;" src="420v21n01-90385615fig3.jpg"></img></p><p class="elsevierStylePara"> Figure 3&#46; High-resolution chest tomography in patient no&#46; 2 shows a pleural thickening with associated subpleural fibrosis&#44; mainly in the upper lobes&#46;</p><p class="elsevierStylePara"><a name="f0020" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img src="420v21n01-90385615fig4.jpg"></img></p><p class="elsevierStylePara"> Figure 4&#46; Histological features of patient no&#46; 2 shows the sharped transition between normal lung parenchyma and the fibroelastotic lesion&#59; orcein elastic stain marks the alveolar walls elastosis &#40;&#916;&#41; and the intra-alveolar fibrosis &#40;<a name="1" class="elsevierStyleCrossRefs"></a><img src="420v21n01-90385615figfx1.jpg"></img> &#41; &#40;H&#38;E&#44; 40&#215;&#44; 100&#215;&#41;&#46;</p><p class="elsevierStylePara"><a name="sec0020" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Discussion</span><p class="elsevierStylePara"> This report describes two cases in which clinical presentation&#44; imaging&#44; and histopathological features are compatible with IPPFE&#46; The recent update of the international multidisciplinary classification of the IIPs recognizes IPPFE as a specific rare IIP&#46;<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a></p><p class="elsevierStylePara"> A review of the available data shows that these two patients were older than the reported median age of 57 years at diagnosis&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> However&#44; the absence of smoking habits is in line with the previous reports&#46;<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a></p><p class="elsevierStylePara"> There is little information and insufficient knowledge regarding the etiology of IPPFE and most cases are considered idiopathic&#44; although a few cases have underlying diseases or conditions such as collagen vascular diseases&#44; bone-marrow transplantation&#44;<a href="&#35;bib4" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">4</span></a> or lung transplantation&#46;<a href="&#35;bib7" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">7</span></a> Genetic predisposition is probably another factor&#46; Frankel et al<span class="elsevierStyleItalic">&#46;</span> reported two cases believed to have familial&#47;genetic lung disease&#46;<a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> Reddy et al<span class="elsevierStyleItalic">&#46;</span> reported that just over half of the patients studied had recurrent lower respiratory tract infections and speculated that repeated inflammatory damage in a predisposed individual may lead to a pattern of intra-alveolar fibrosis with septal elastosis &#40;IAFE&#41;&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> In fact&#44; the second case described in this manuscript has a history of recurrent respiratory infections&#46;</p><p class="elsevierStylePara"> There is a certain variability of clinical presentation among the reported IPPFE patients&#44; such as spontaneous pneumothorax&#44; dyspnea&#44; or chronic cough&#46; However&#44; in a recently published series of clinical cases&#44;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> the most frequent presentation symptoms were shortness of breath in 91&#46;7&#37; and dry cough in 50&#37; of the patients&#44; which fit in with the complaints mentioned by the patients reported in this paper&#46;</p><p class="elsevierStylePara"> Computed tomography findings in both cases revealed bilateral pleuroparenchymal thickening&#44; which was mostly marked in the upper zones&#44; with an associated subpleural reticular pattern consistent with fibrosis&#46; These findings are also consistent with previous series described in literature&#46;<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a></p><p class="elsevierStylePara"> In both cases&#44; CT-guided transthoracic lung biopsies were made in upper lobes and demonstrated IAFE&#46; The transition between the fibroelastosis and the underlying normal lung parenchyma was abrupt&#46; However&#44; there is no standard histological criterion for the diagnosis of IPPFE&#46; Reddy et al&#46;&#44; using published histological criteria&#44; characterized them as &#8220;definite&#8221; when there was upper zone pleural fibrosis with subjacent intra-alveolar fibrosis accompanied by alveolar septal elastosis&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> Kusagaya et al<span class="elsevierStyleItalic">&#46;</span> used the pathological criteria for the diagnosis of IPPFE as follows&#58; intense fibrosis of the visceral pleura&#59; prominent&#44; homogeneous&#44; subpleural fibroelastosis&#59; sparing of the parenchyma distant from the pleura&#59; mild&#44; patchy lymphoplasmocytic infiltrates&#44; and presence of small numbers of fibroblastic foci&#46;<a href="&#35;bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">8</span></a> IAFE is not specific to IPPFE&#44; being a pathway of lung injury common to a variety of disorders&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> A multidisciplinary approach&#44; integrating all clinical&#44; imaging and histopathological findings led to an IPPFE diagnosis in both cases&#46;</p><p class="elsevierStylePara"> Published case series<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">8</span></a> have included patients who were submitted to surgical lung biopsy&#46; However&#44; considering that the two described cases were already complicated by iatrogenic pneumothorax&#44; the risk of performing this procedure was very high&#46; Becker et al&#46; postulate that these patients may be prone to the development of secondary spontaneous pneumothorax and reported a death following a surgical lung biopsy complicated by a large bronchopleural fistulae&#46;<a href="&#35;bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">9</span></a> Since the histological features obtained by CT-guided transthoracic core lung biopsy were considered representative and compatible with the diagnosis of IPPFE &#40;after exclusion&#44; in a multidisciplinary discussion&#44; of apical cap&#44; the most frequent differential diagnosis&#41;&#44; it was decided not to carry out a surgical biopsy&#46;</p><p class="elsevierStylePara"> There is no defined treatment and the therapeutic approach varies greatly and is largely empirical in the published clinical cases&#44; with some of them reporting aggressive treatment with corticosteroids and immunosupressants&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> Based on this report&#44; we also considered this therapeutic approach with the first patient diagnosed with IPPFE&#46; However&#44; since the hypothesis of recurrent respiratory infections as a possible etiology of this disease had been described and increasingly considered likely&#44; we decided on a different option for the second patient with the prescription of hydroxychloroquine and a low dose of corticosteroids&#46;</p><p class="elsevierStylePara"> There is more recognition of the etiology and physiopathology of IPPFE&#44; however it would be important for us to know more about the efficacy of different therapeutics administered so far and the evolution of the patients&#46;</p><p class="elsevierStylePara"><a name="sec0025" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Conflict of interest</span><p class="elsevierStylePara"> The authors have no conflicts of interest to declare&#46;</p><p class="elsevierStylePara"> Received 3 December 2013 <br></br> Accepted 29 April 2014</p><p class="elsevierStylePara"> Corresponding author&#46; margarida&#46;tredondo&#64;gmail&#46;com</p>"
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        "resumen" => "<span class="elsevierStyleSectionTitle"> Abstract</span><br/><p class="elsevierStylePara"> Idiopathic pleuroparenchymal fibroelastosis &#40;IPPFE&#41; is a recently described rare entity&#44; characterized by pleural and subpleural parenchymal fibrosis and elastosis mainly in the upper lobes&#46; The etiology and pathophysiology are unknown&#46; The prognosis is poor&#44; with no effective therapies other than lung transplantation&#46; IPPFE should be properly identified so that it can be approached correctly&#46; This report describes two clinical cases with clinical imaging and histological features compatible with IPPFE&#46;</p>"
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Idiopathic pleuroparenchymal fibroelastosis: A rare but increasingly recognized entity
M.T.. Redondoa,
Corresponding author
margarida.tredondo@gmail.com

Corresponding author. margarida.tredondo@gmail.com
, N.. Meloa, P.C.. Motaa,b, J.M.. Jesusc, C.S.. Mourab,d, S.. Guimarãesd, A.. Moraisa,b
a Department of Pneumology, Centro Hospitalar de São João, Portugal
b Faculty of Medicine of University of Porto, Portugal
c Department of Radiology, Centro Hospitalar de São João, Portugal
d Department of Pathology, Centro Hospitalar de São João, Portugal
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    "textoCompleto" => "<p class="elsevierStylePara"><a name="sec0005" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Introduction</span><p class="elsevierStylePara"> Idiopathic pleuroparenchymal fibroelastosis &#40;IPPFE&#41; is a rare condition characterized by fibroelastotic thickening of the pleural and subpleural lung parenchyma&#44; mainly in the upper lobes&#46;<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a> This entity was first mentioned in English-language medical literature by Frankel et al&#46;<a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> in 2004&#44; although the same concept designated as idiopathic pulmonary upper lobe fibrosis was proposed in 1992 by Aminati et al&#46;<a href="&#35;bib3" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> There is little known about its etiology and most cases are considered to be idiopathic&#44; although a few cases are believed to occur in a familial&#47;genetic lung disease context<a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> and others have been reported in association with previous bone marrow transplantation&#46;<a href="&#35;bib4" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">4</span></a> New data have been published suggesting that recurrent infections may have a role in its pathogenesis&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> Recently IPPFE was recognized as a specific rare idiopathic interstitial pneumonia &#40;IIP&#41; in the update of the international multidisciplinary classification of the IIPs<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a>&#59; IPPFE was characterized by an elastotic fibrosis present with an intra-alveolar fibrosis&#46;<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a> High-resolution chest tomography &#40;HRCT&#41; shows dense subpleural consolidation with traction bronchiectasis&#44; architectural distortion&#44; and upper lobe volume loss&#46;<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a> The authors present two cases of IPPFE in which the final diagnosis was obtained after CT-guided transthoracic core lung biopsy&#46;</p><p class="elsevierStylePara"><a name="sec0010" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Case report 1</span><p class="elsevierStylePara"> The patient was a 66-year-old female&#44; caucasian&#44; nonsmoker&#44; had worked as art gallery assistant&#44; had a history of esophageal hiatus hernia and was chronically medicated with omeprazole&#46; She had no known family history of pulmonary disease&#46; In September 2010&#44; she complained of dyspnea on exertion and nonproductive cough&#44; with progressive worsening&#46; The patient was examined by a general practitioner and a chest X-ray was performed that revealed a reticular densification with peripheral distribution and predominance in the upper lobes&#46; Lung function tests were as follows&#58; FVC 1&#46;74&#160;L &#40;79&#46;4&#37; predicted&#41;&#44; FEV1 1&#46;51&#160;L &#40;83&#46;5&#37; predicted&#41;&#44; FEV1&#47;FVC 86&#46;8&#37;&#44; TLC 4&#46;16&#160;L &#40;95&#46;2&#37; predicted&#41;&#44; DLco 2&#46;85&#160;mL&#47;min&#47;mmHg &#40;44&#46;6&#37; predicted&#41;&#46; Blood gas parameters were in the reference range values&#46; She walked 450&#160;m at 6-min walk test &#40;6-WT&#41; with an oxygen desaturation of 12&#37; &#40;initial SatO2-97&#37; and final SatO2-85&#37;&#41;&#46; HRCT images showed pleural and subpleural thickening with fibrotic changes in the marginal parenchyma&#44; mainly in the upper lobes &#40;<a href="&#35;f0005" class="elsevierStyleCrossRefs">Figure 1</a>&#41;&#46; A short course of corticosteroids was prescribed with no clinical improvement&#46; The patient was then referred to the Interstitial Lung Disease outpatient clinic&#46; On chest examination&#44; fine bibasilar inspiratory rales were identified&#46; Autoimmune serological testing was negative&#46; A bronchoscopy was performed with no noticeable airways abnormalities&#46; Bronchoalveolar lavage revealed a lymphocytosis &#40;28&#46;4&#37;&#41; with a very high CD4&#47;CD8 ratio &#40;14&#46;5&#41;&#44; and no microorganism or malignant cells were identified&#46; Transbronchial and bronchial biopsies had no evidence of malignancy or granulomas&#46; The patient underwent a CT-guided transthoracic core lung biopsy in two distinct locations&#44; complicated by pneumothorax with bronchopleural fistula and subcutaneous emphysema&#46; Histological evaluation showed marked thickened visceral pleura and prominent predominantly elastic sub-pleural fibrosis with mild&#44; patchy lymphoplasmacytic inflammatory infiltrate&#59; orcein stain showed that the elastosis was within the alveolar walls&#44; marking the intra-alveolar fibrosis &#40;<a href="&#35;f0010" class="elsevierStyleCrossRefs">Figure 2</a>&#41;&#46; Despite the prescription of azathioprine &#40;2&#160;mg&#47;kg&#47;day&#41;&#44; the patient is clinically and functionally worse after 20 months of follow-up&#58; FVC 1&#46;63&#160;L &#40;82&#37; predicted&#41;&#44; FEV1 1&#46;44&#160;L &#40;89&#37; predicted&#41;&#44; FEV1&#47;FVC 89&#46;5&#37;&#44; TLC 3&#46;28&#160;L &#40;80&#37; predicted&#41;&#44; DLco 1&#46;66&#160;mL&#47;min&#47;mmHg &#40;26&#37; predicted&#41;&#46;</p><p class="elsevierStylePara"><a name="f0005" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img alt="High-resolution chest tomography in patient no&#46; 1 shows pleural and subpleural thickening with fibrotic changes in the marginal parenchyma&#44; mainly in the upper lobes&#46;" src="420v21n01-90385615fig1.jpg"></img></p><p class="elsevierStylePara"> Figure 1&#46; High-resolution chest tomography in patient no&#46; 1 shows pleural and subpleural thickening with fibrotic changes in the marginal parenchyma&#44; mainly in the upper lobes&#46;</p><p class="elsevierStylePara"><a name="f0010" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img src="420v21n01-90385615fig2.jpg"></img></p><p class="elsevierStylePara"> Figure 2&#46; Histological features of patient no&#46; 1 shows a sub-pleural elastotic fibrosis highlighted by the orcein elastic stain&#44; that marks the alveolar walls elastosis &#40;&#916;&#41; and the intra-alveolar fibrosis &#40;<a name="0" class="elsevierStyleCrossRefs"></a><img src="420v21n01-90385615figfx1.jpg"></img> &#41; &#40;H&#38;E&#44; 100&#215;&#59; Orcein&#44; 40&#215;&#41;&#46;</p><p class="elsevierStylePara"><a name="sec0015" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Case report 2</span><p class="elsevierStylePara"> The patient was a 59-year-old female&#44; caucasian&#44; nonsmoker&#44; accountant&#44; with history of pulmonary tuberculosis &#40;5-years-old&#41;&#44; post-infectious bronchiectasis&#44; recurrent respiratory infections&#44; and osteoporosis&#46; The patient had no known family history of pulmonary disease&#46; Since the age of 50 she had attended a respiratory outpatient clinic because of dyspnea on exertion and pleural thickening in the upper lobes&#46; These findings were interpreted as within the clinical picture of sequelae of tuberculosis and the patient was medicated with an inhaled association of budesonide and formoterol&#46; However&#44; after 7 years of follow-up&#44; the patient presented a worsening of dyspnea on exertion and dry cough&#46; On chest examination&#44; fine bibasilar inspiratory rales were identified&#46; Chest X-ray revealed a linear densification with peripheral distribution and predominance in the upper lobes&#44; with significant worsening compared to previous ones&#46; HRCT showed a pleural thickening with associated subpleural fibrosis&#44; mainly in the upper lobes &#40;<a href="&#35;f0015" class="elsevierStyleCrossRefs">Figure 3</a>&#41;&#46; Autoimmune serological testing was negative&#46; Lung function tests showed moderate restrictive ventilatory impairment and a significant decrease in diffusion capacity&#58; FVC 1&#46;37&#160;L &#40;56&#46;1&#37; predicted&#41;&#44; FEV1 1&#46;17&#160;L &#40;57&#46;3&#37; predicted&#41;&#44; FEV1&#47;FVC 85&#46;5&#37;&#44; TLC 2&#46;87&#160;L &#40;64&#46;7&#37; predicted&#41;&#44; DLco 2&#46;07&#160;mL&#47;min&#47;mmHg &#40;29&#46;4&#37; predicted&#41;&#46; Blood gas parameters were in the reference range values&#46; She walked 435&#160;m at 6-WT with an oxygen desaturation of 10&#37; &#40;initial SatO2-96&#37; and final SatO2-86&#37;&#41;&#46; A bronchoscopy was performed with no noticeable airways abnormalities&#46; Bronchoalveolar lavage revealed neutrophilic &#40;53&#46;8&#37;&#41; and eosinophilic &#40;14&#37;&#41; alveolitis and no microorganism or malignant cells were identified&#46; CT-guided transthoracic core lung biopsy was then performed with iatrogenic pneumothorax&#46; Histological evaluation showed marked thickened visceral pleura and sharply demarcated elastic fibrosis&#44; without significant inflammation&#59; orcein stain showed elastosis of the alveolar walls with a predominant intra-alveolar fibrosis &#40;<a href="&#35;f0020" class="elsevierStyleCrossRefs">Figure 4</a>&#41;&#46; Treatment was started with 400&#160;mg&#47;day of hydroxychloroquine and 7&#46;5&#160;mg&#47;day of prednisolone&#46; At the time of writing&#44; after a 10-month follow-up period&#44; the patient is clinically and functionally stable&#58; FVC 1&#46;27&#160;L &#40;54&#37; predicted&#41;&#44; FEV1 1&#46;07&#160;L &#40;54&#37; predicted&#41;&#44; FEV1&#47;FVC 84&#46;4&#37;&#44; TLC 2&#46;94&#160;L &#40;68&#37; predicted&#41;&#44; DLco 1&#46;52&#160;mL&#47;min&#47;mmHg &#40;22&#37; predicted&#41;&#46;</p><p class="elsevierStylePara"><a name="f0015" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img alt="High-resolution chest tomography in patient no&#46; 2 shows a pleural thickening with associated subpleural fibrosis&#44; mainly in the upper lobes&#46;" src="420v21n01-90385615fig3.jpg"></img></p><p class="elsevierStylePara"> Figure 3&#46; High-resolution chest tomography in patient no&#46; 2 shows a pleural thickening with associated subpleural fibrosis&#44; mainly in the upper lobes&#46;</p><p class="elsevierStylePara"><a name="f0020" class="elsevierStyleCrossRefs"></a></p><p class="elsevierStylePara"><img src="420v21n01-90385615fig4.jpg"></img></p><p class="elsevierStylePara"> Figure 4&#46; Histological features of patient no&#46; 2 shows the sharped transition between normal lung parenchyma and the fibroelastotic lesion&#59; orcein elastic stain marks the alveolar walls elastosis &#40;&#916;&#41; and the intra-alveolar fibrosis &#40;<a name="1" class="elsevierStyleCrossRefs"></a><img src="420v21n01-90385615figfx1.jpg"></img> &#41; &#40;H&#38;E&#44; 40&#215;&#44; 100&#215;&#41;&#46;</p><p class="elsevierStylePara"><a name="sec0020" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Discussion</span><p class="elsevierStylePara"> This report describes two cases in which clinical presentation&#44; imaging&#44; and histopathological features are compatible with IPPFE&#46; The recent update of the international multidisciplinary classification of the IIPs recognizes IPPFE as a specific rare IIP&#46;<a href="&#35;bib6" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a></p><p class="elsevierStylePara"> A review of the available data shows that these two patients were older than the reported median age of 57 years at diagnosis&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> However&#44; the absence of smoking habits is in line with the previous reports&#46;<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a></p><p class="elsevierStylePara"> There is little information and insufficient knowledge regarding the etiology of IPPFE and most cases are considered idiopathic&#44; although a few cases have underlying diseases or conditions such as collagen vascular diseases&#44; bone-marrow transplantation&#44;<a href="&#35;bib4" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">4</span></a> or lung transplantation&#46;<a href="&#35;bib7" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">7</span></a> Genetic predisposition is probably another factor&#46; Frankel et al<span class="elsevierStyleItalic">&#46;</span> reported two cases believed to have familial&#47;genetic lung disease&#46;<a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> Reddy et al<span class="elsevierStyleItalic">&#46;</span> reported that just over half of the patients studied had recurrent lower respiratory tract infections and speculated that repeated inflammatory damage in a predisposed individual may lead to a pattern of intra-alveolar fibrosis with septal elastosis &#40;IAFE&#41;&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> In fact&#44; the second case described in this manuscript has a history of recurrent respiratory infections&#46;</p><p class="elsevierStylePara"> There is a certain variability of clinical presentation among the reported IPPFE patients&#44; such as spontaneous pneumothorax&#44; dyspnea&#44; or chronic cough&#46; However&#44; in a recently published series of clinical cases&#44;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> the most frequent presentation symptoms were shortness of breath in 91&#46;7&#37; and dry cough in 50&#37; of the patients&#44; which fit in with the complaints mentioned by the patients reported in this paper&#46;</p><p class="elsevierStylePara"> Computed tomography findings in both cases revealed bilateral pleuroparenchymal thickening&#44; which was mostly marked in the upper zones&#44; with an associated subpleural reticular pattern consistent with fibrosis&#46; These findings are also consistent with previous series described in literature&#46;<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib2" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a></p><p class="elsevierStylePara"> In both cases&#44; CT-guided transthoracic lung biopsies were made in upper lobes and demonstrated IAFE&#46; The transition between the fibroelastosis and the underlying normal lung parenchyma was abrupt&#46; However&#44; there is no standard histological criterion for the diagnosis of IPPFE&#46; Reddy et al&#46;&#44; using published histological criteria&#44; characterized them as &#8220;definite&#8221; when there was upper zone pleural fibrosis with subjacent intra-alveolar fibrosis accompanied by alveolar septal elastosis&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> Kusagaya et al<span class="elsevierStyleItalic">&#46;</span> used the pathological criteria for the diagnosis of IPPFE as follows&#58; intense fibrosis of the visceral pleura&#59; prominent&#44; homogeneous&#44; subpleural fibroelastosis&#59; sparing of the parenchyma distant from the pleura&#59; mild&#44; patchy lymphoplasmocytic infiltrates&#44; and presence of small numbers of fibroblastic foci&#46;<a href="&#35;bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">8</span></a> IAFE is not specific to IPPFE&#44; being a pathway of lung injury common to a variety of disorders&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> A multidisciplinary approach&#44; integrating all clinical&#44; imaging and histopathological findings led to an IPPFE diagnosis in both cases&#46;</p><p class="elsevierStylePara"> Published case series<a href="&#35;bib1" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a><span class="elsevierStyleSup">&#44; </span><a href="&#35;bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">8</span></a> have included patients who were submitted to surgical lung biopsy&#46; However&#44; considering that the two described cases were already complicated by iatrogenic pneumothorax&#44; the risk of performing this procedure was very high&#46; Becker et al&#46; postulate that these patients may be prone to the development of secondary spontaneous pneumothorax and reported a death following a surgical lung biopsy complicated by a large bronchopleural fistulae&#46;<a href="&#35;bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">9</span></a> Since the histological features obtained by CT-guided transthoracic core lung biopsy were considered representative and compatible with the diagnosis of IPPFE &#40;after exclusion&#44; in a multidisciplinary discussion&#44; of apical cap&#44; the most frequent differential diagnosis&#41;&#44; it was decided not to carry out a surgical biopsy&#46;</p><p class="elsevierStylePara"> There is no defined treatment and the therapeutic approach varies greatly and is largely empirical in the published clinical cases&#44; with some of them reporting aggressive treatment with corticosteroids and immunosupressants&#46;<a href="&#35;bib5" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> Based on this report&#44; we also considered this therapeutic approach with the first patient diagnosed with IPPFE&#46; However&#44; since the hypothesis of recurrent respiratory infections as a possible etiology of this disease had been described and increasingly considered likely&#44; we decided on a different option for the second patient with the prescription of hydroxychloroquine and a low dose of corticosteroids&#46;</p><p class="elsevierStylePara"> There is more recognition of the etiology and physiopathology of IPPFE&#44; however it would be important for us to know more about the efficacy of different therapeutics administered so far and the evolution of the patients&#46;</p><p class="elsevierStylePara"><a name="sec0025" class="elsevierStyleCrossRefs"></a></p><span class="elsevierStyleSectionTitle"> Conflict of interest</span><p class="elsevierStylePara"> The authors have no conflicts of interest to declare&#46;</p><p class="elsevierStylePara"> Received 3 December 2013 <br></br> Accepted 29 April 2014</p><p class="elsevierStylePara"> Corresponding author&#46; margarida&#46;tredondo&#64;gmail&#46;com</p>"
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        "resumen" => "<span class="elsevierStyleSectionTitle"> Abstract</span><br/><p class="elsevierStylePara"> Idiopathic pleuroparenchymal fibroelastosis &#40;IPPFE&#41; is a recently described rare entity&#44; characterized by pleural and subpleural parenchymal fibrosis and elastosis mainly in the upper lobes&#46; The etiology and pathophysiology are unknown&#46; The prognosis is poor&#44; with no effective therapies other than lung transplantation&#46; IPPFE should be properly identified so that it can be approached correctly&#46; This report describes two clinical cases with clinical imaging and histological features compatible with IPPFE&#46;</p>"
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Article information
ISSN: 08732159
Original language: English
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2021 November 78 32 110
2021 October 90 57 147
2021 September 97 27 124
2021 August 96 36 132
2021 July 88 19 107
2021 June 116 32 148
2021 May 89 24 113
2021 April 221 62 283
2021 March 184 31 215
2021 February 115 21 136
2021 January 73 19 92
2020 December 67 14 81
2020 November 141 21 162
2020 October 98 23 121
2020 September 141 34 175
2020 August 119 21 140
2020 July 153 22 175
2020 June 161 21 182
2020 May 139 22 161
2020 April 133 17 150
2020 March 130 9 139
2020 February 187 25 212
2020 January 139 24 163
2019 December 115 17 132
2019 November 154 20 174
2019 October 142 33 175
2019 September 149 33 182
2019 August 245 21 266
2019 July 244 26 270
2019 June 238 8 246
2019 May 254 28 282
2019 April 193 22 215
2019 March 302 9 311
2019 February 233 12 245
2019 January 261 24 285
2018 December 113 4 117
2018 November 39 0 39
2018 October 47 12 59
2018 September 24 6 30
2018 August 38 27 65
2018 July 40 16 56
2018 June 32 17 49
2018 May 53 18 71
2018 April 78 19 97
2018 March 70 16 86
2018 February 53 15 68
2018 January 55 11 66
2017 December 78 18 96
2017 November 33 10 43
2017 October 15 15 30
2017 September 12 12 24
2017 August 18 10 28
2017 July 12 17 29
2017 June 23 17 40
2017 May 22 15 37
2017 April 9 5 14
2017 March 15 4 19
2017 February 8 4 12
2017 January 8 11 19
2016 December 10 6 16
2016 November 10 2 12
2016 October 17 9 26
2016 September 3 3 6
2016 August 9 6 15
2016 July 4 7 11
2016 June 5 5 10
2016 May 20 4 24
2016 April 98 2 100
2016 March 173 53 226
2016 February 167 39 206
2016 January 148 40 188
2015 December 147 33 180
2015 November 128 27 155
2015 October 135 64 199
2015 September 114 31 145
2015 August 114 25 139
2015 July 144 11 155
2015 June 66 12 78
2015 May 76 24 100
2015 April 102 36 138
2015 March 111 41 152
2015 February 82 67 149
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