Desquamative interstitial pneumonia: a systematic review of its features and outcomes

Hellemons, Merel E.; Moor, Catharina C.; Thüsen, Jan von der; Rossius, Mariska; Odink, Arlette E.; Thorgersen, Laila Haugen; Verschakelen, Johny; Wuyts, Wim; Wijsenbeek, Marlies; Bendstrup, Elisabeth

doi:10.1183/16000617.0181-2019

Cited by 38 publications

(41 citation statements)

References 80 publications

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“…La neumonía intersticial descamativa (NID) es un padecimiento raro: solamente se han reportado 362 casos en adultos, la mayoría entre fumadores [ 1 ]. En niños, no había consenso sobre su etiología y mecanismo patológico en los 41 artículos escritos en inglés hallados en la literatura hasta 2020 [ 1 , 2 , 3 ]. Hasta donde sabemos, solamente tres de los casos de NID reportados en niños presentaron insuficiencia renal asociada [ 4 ], y ninguno mostró un deterioro rápido y fatal en pacientes menores de 2 años.…”

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Hallazgos similares al COVID-19 en un caso fatal de neumonía intersticial descamativa asociada con glomerulonefritis por IgA en una niña de 13 meses de edad

et al. 2021

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En la era del COVID-19, es usual sospechar que cualquier paciente con síndrome respiratorio agudo grave (SARS) esté asociado con una infección por SARS-CoV-2. El objetivo de este artículo es presentar un caso de neumonía similar al COVID, con evolución fatal. Los aspectos clínicos se correlacionan con los hallazgos en la autopsia y se discuten en el contexto de los datos más recientes en la literatura médica. Una niña de 13 meses de edad ingresó a la sala de emergencias con dificultad respiratoria aguda y opacidades pulmonares bilaterales con apariencia de vidrio molido, además del pulmón izquierdo casi completamente opacificado. El estado de la paciente se deterioró súbitamente, y se confirmó la muerte 3 h después de la admisión. En la autopsia se diagnosticó neumonía intersticial descamativa grave, y se asoció con glomerulonefritis por IgA, un hallazgo poco usual. No se detectó infección por SARS-CoV-2 en el parénquima pulmonar mediante RT-PCR. Éste es un caso muy inusual de deterioro rápido de un infante con neumonía intersticial descamativa (NID) idiopática y afectación multiorgánica. Con base en tinciones inmunohistoquímicas, proponemos la hipótesis de que, en la NID, las membranas hialinas surgen de neumocitos descamados necrotizantes. En la era de COVID-19, tales casos son extremadamente difíciles de diagnosticar, y pueden semejar las lesiones pulmonares inducidas por el SARS-CoV-2. Esta pauta de formación de membrana hialina podría explicar la falta de respuesta a la terapia con oxígeno. El presente caso resalta la importancia de la autopsia en estos casos complicados.

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Section: Introductionunclassified

Hallazgos similares al COVID-19 en un caso fatal de neumonía intersticial descamativa asociada con glomerulonefritis por IgA en una niña de 13 meses de edad

et al. 2021

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“…Desquamative interstitial pneumonia (DIP) is a rare disorder: only 362 cases have been reported in adults, mostly among smokers (1). In children, its etiology and pathomechanism were far from reaching consensus in the 41 English-language articles found in the literature until 2020 (1)(2)(3). To the best of our knowledge, only three of the reported DIP cases in children presented with associated kidney failure (4), and not one featured rapid and fatal deterioration in patients below 2 years old.…”

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confidence: 99%

COVID-19 Like Findings in a Fatal Case of Idiopathic Desquamative Interstitial Pneumonia Associated With IgA Glomerulonephritis in a 13-Month-Old Child

et al. 2020

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In the COVID-19 era, patients with severe acute respiratory syndrome (SARS) are suspected to be associated with SARS-CoV-2 infection. The aim of this paper is to present a case with COVID-like pneumonia, with fatal evolution. The clinical aspects were correlated with the autopsy findings and discussed on the background of the most recent data from the medical literature. A 13-month-old girl was admitted to the emergency room with acute severe shortness of breath and pulmonary bilateral ground-glass opacities and an almost complete opacified left lung. The patient suddenly deteriorated, and death was confirmed 3 h after admission. At autopsy, severe desquamative interstitial pneumonia was diagnosed and was associated with an unusual IgA glomerulonephritis. No SARS-CoV-2 infection was detected in the lung parenchyma by RT- PCR. This is a very unusual case of rapid deterioration of an infant with idiopathic desquamative interstitial pneumonia (IDP) and multiorgan involvement. Based on immunohistochemical stains, we hypothesize that, in IDP, the hyaline membranes arise from necrotizing desquamated pneumocytes. In the COVID-19 era, such cases are extremely difficult to diagnose; they can mimic SARS-CoV-2-induced lung injuries. This pattern of hyaline membrane formation might explain the poor response to oxygen therapy. The present case highlights the importance of autopsy in such challenging cases.

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“…Publications of series are scarce and spread over many years, limiting access to synthetic information. In the current issue of the European Respiratory Review, HELLEMONS et al [16] performed a systematic literature search of DIP, and reviewed a total of 362 cases of DIP from 13 case series and 68 individual cases in adults published between 1965 and 2019. They reported dyspnoea and cough as the most common presenting symptoms, and presence of a restrictive physiology in 71% of cases with reduced diffusion capacity.…”

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confidence: 99%

“…While these results were not surprising, the systematic review provides us with a much more accurate overview than previously available from individual cases or small series. Furthermore, HELLEMONS et al [16] identified data regarding treatment received in a majority of cases. Removal of the suspected culprit and initiation of glucocorticoids were the most frequent treatments.…”

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confidence: 99%

“…Whether smoking cessation indeed affects the outcome of DIP has not been demonstrated. More than half of the patients with DIP who receive glucocorticoids seem to benefit from therapy [16][17][18][19] and have a relatively good prognosis [10,16]; however, progression to chronic respiratory failure, need for transplantation or death were also reported [20,21]. Among patients with a known smoking history, 70% of patients who had smoked had a stable course of disease or improved; in contrast, only 40% of those without a history of smoking improved or remained stable and the majority experienced disease worsening (especially in women) [16].…”

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Desquamative interstitial pneumonia: still orphan and not always benign

Cottin

2020

Eur Respir Rev

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Desquamative interstitial pneumonia (DIP) was originally described by LIEBOW et al. [1] in 1965, and so named because of the observation of cells filling the alveolar spaces and the belief that this feature was due to desquamation of alveolar epithelial cells. It has since been recognised that the dominant histologic feature of DIP represents accumulation of intra-alveolar macrophages, and sometimes of giant cells [2]. Although the more accurate terminology of "alveolar macrophage pneumonia" has been proposed, the term DIP has persisted [3].In the international classification of idiopathic interstitial pneumonias [3], DIP and respiratory bronchiolitis-interstitial lung disease (RB-ILD) belong to the group of smoking-related interstitial pneumonia, together with pulmonary Langerhans cell granulomatosis. Depending on the classification, combined pulmonary fibrosis and emphysema, smoking-related acute interstitial pneumonia, smoking-related interstitial fibrosis, rheumatoid arthritis-associated ILD, and idiopathic pulmonary fibrosis may also be considered as smoking-related ILDs [4,5]. DIP and RB-ILD are part of a histologic spectrum of macrophage accumulation, with the distinction dependent on the extent and distribution of this process. Histologic distinction between DIP and RB-ILD is sometimes difficult, hence the terminology of smoking-induced ILD [6]. RB, a finding present in the lung of almost all smokers, is characterised by the deposition of tobacco pigmented-macrophages predominating in the respiratory bronchioles and peribronchiolar alveolar spaces [7]. RB-ILD is a form of interstitial pneumonia, where RB is associated with a chronic ILD predominating in peribronchial areas and resulting from extensive response to smoking, with pathologic, clinical, and radiologic features [8]. DIP is characterised by prominent, diffuse intra-alveolar accumulation of pigmented macrophages, hyperplasia of type II alveolar epithelial cells, and often diffuse alveolar septal thickening, with possible septal fibrosis and mild interstitial inflammation [1,6,9]. In addition to demonstrating a large number of pigmented macrophages, bronchoalveolar lavage may show an increased number of eosinophils [10], but whether eosinophils are associated with a different response to glucocorticoids or a different outcome is unclear.Distinctions between DIP and RB-ILD also reflect in the pattern of disease on high-resolution computed tomography [11], with centrilobular nodules and ground-glass attenuation predominating in the upper

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Desquamative interstitial pneumonia: a systematic review of its features and outcomes

Cited by 38 publications

References 80 publications

Hallazgos similares al COVID-19 en un caso fatal de neumonía intersticial descamativa asociada con glomerulonefritis por IgA en una niña de 13 meses de edad

Hallazgos similares al COVID-19 en un caso fatal de neumonía intersticial descamativa asociada con glomerulonefritis por IgA en una niña de 13 meses de edad

COVID-19 Like Findings in a Fatal Case of Idiopathic Desquamative Interstitial Pneumonia Associated With IgA Glomerulonephritis in a 13-Month-Old Child

Desquamative interstitial pneumonia: still orphan and not always benign

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