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

Merel E. Hellemons, Catharina C. Moor, Jan von der Thüsen, Mariska Rossius, Arlette Odink, Laila Haugen Thorgersen, Johny Verschakelen, Wim Wuyts, Marlies S. Wijsenbeek, Elisabeth Bendstrup
European Respiratory Review 2020 29: 190181; DOI: 10.1183/16000617.0181-2019
Merel E. Hellemons
1Dept of Interstitial Lung Disease, Erasmus Medical Center, Rotterdam, The Netherlands
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
9These authors share first authorship
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Catharina C. Moor
1Dept of Interstitial Lung Disease, Erasmus Medical Center, Rotterdam, The Netherlands
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
9These authors share first authorship
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Jan von der Thüsen
2Dept of Pathology, Erasmus Medical Center, Rotterdam, The Netherlands
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
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Mariska Rossius
3Dept of Radiology and Nuclear Medicine, Erasmus Medical Center, Rotterdam, The Netherlands
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
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Arlette Odink
3Dept of Radiology and Nuclear Medicine, Erasmus Medical Center, Rotterdam, The Netherlands
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
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Laila Haugen Thorgersen
4Dept of Respiratory Diseases, Nastved Hospital, Nastved, Denmark
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Johny Verschakelen
5Dept of Radiology, University Hospital Leuven, Leuven, Belgium
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
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Wim Wuyts
6Unit of Interstitial Lung Disease, University Hospital Leuven, Leuven, Belgium
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
10These authors share senior authorship
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  • ORCID record for Wim Wuyts
Marlies S. Wijsenbeek
1Dept of Interstitial Lung Disease, Erasmus Medical Center, Rotterdam, The Netherlands
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
10These authors share senior authorship
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  • For correspondence: m.wijsenbeek-lourens@erasmusmc.nl
Elisabeth Bendstrup
7Dept of Respiratory Diseases and Allergy, Center for Rare Lung Diseases, Aarhus University Hospital, Aarhus, Denmark
8This centre is a member of the European Reference Network for Rare Lung Diseases/Interstitial Lung Diseases (ERN-LUNG ILD)
10These authors share senior authorship
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  • FIGURE 1
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    FIGURE 1

    Typical example of high-resolution computed tomography (HRCT) findings in desquamative interstitial pneumonia (DIP). Coronal a) and axial b) computed tomography (CT) slices show bilateral basal and peripheral ground-glass opacity. Some superimposed fine linear opacities are also seen, especially on the left side (coronal slice a)), corresponding with thickened inter- and intralobular septa.

  • FIGURE 2
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    FIGURE 2

    Typical examples of histopathological findings in desquamative interstitial pneumonia (DIP) using a hematoxylin–eosin stain. The low-power view a) demonstrates diffuse filling of alveolar spaces with macrophages, as well as diffuse widening of alveolar septa (asterisks) suggestive of a fibrotic non-specific interstitial pneumonia (NSIP) pattern. Scale bar=1 mm. The medium-power view b) shows complete filling of a single alveolus with slightly pigmented macrophages (asterisk). Scale bar=0.1 mm. The high-power view c) shows reactive pneumocytic epithelial cells, alveolar filling with macrophages and sporadic eosinophils (arrows). Scale bar=0.05 mm.

Tables

  • Figures
  • Supplementary Materials
  • TABLE 1

    Patient demographics and clinical characteristics

    Study (first author)LiebowGaenslerTubbsValdiviaCarringtonYousemHartmanAkiraTravisRyuCraigBaloiraKawabataIndividual casesTotal
    Country of studyUSAUSAUSAUSAUSAUSACanadaJapanUSAUSAUKSpainJapanVarious–
    Year published19651966197719771978198919931997200020042004200720121966–2019–
    Patients181226304036228162320123168362
    Male gender10 (56)7 (58)17 (65)–28 (70)26 (72)6 (22)7 (88)7 (44)11 (48)12 (60)12 (100)29 (96)52 (76)224 (62)
    Age years42 (16–61)39 (17–65)52 (24–75)–42±1242 (17–67)43 (22–71)56±842 (27–79)46±1043 (27–74)50±1655±1345±14–
    Non-productive cough14 (78)–17 (65)––––––10 (43)–9 (75)–46 (68)96/147 (65)
    Exertional dyspnoea18 (100)–23 (88)––––––20 (87)–7 (58)–58 (85)127/147 (86)
    Chest pain9 (50)–1 (4)––––––4 (17)–0 (0)–12 (18)26/147 (19)
    Fever–––––––––––––21 (31)21/68 (31)
    Fatigue–––––––––––––15 (22)15/68 (22)
    Anorexia/weight loss6 (33)–1 (4)––––––––––8 (12)15/112 (13)
    Symptoms prior to presentation months17 (2–72)––––––––––––14 (0.25–120)13 (1–120)
    No symptoms (incidental finding)––1 (4)––––––1 (4)–1 (8)–5 (7)8/129 (6)
    Normal physical examination–––––––––––––13 (19)13/68 (19)
    Crackles––17 (65)––––––13 (57)–11 (92)–41 (72)82/129 (64)
    Clubbing5 (28)–12 (46)––––––6 (26)–7 (58)–18 (32)48/147 (33)

    Data are presented as n, n (%), n/n (%), mean±sd, or mean (range). Available data from case series and individual cases are presented. Totals data are summarised as percentages of available data.

    • TABLE 2

      Patient smoking history

      Study (last name first author)TubbsCarringtonYousemAkiraTravisRyuCraigBaloiraKawabataIndividual casesTotal
      Patients26#40¶368162320123168359
      Smoking status
       Active smoking–––614181242831113 (70)
       No active smoking–––2058822449 (30)
       Former smoking–––102060917 (11)
       Never smoking–––1038221531 (19)
       Unknown26403602000113200 (56)
      Smoking duration pack-years>10>103–38 (3–75)39 (10–90)25 (3–80)–52±4142 (5–150)–

      Data are presented as n, n (%), mean±sd, or mean (range). Available data on smoking exposition in case series and individual cases are presented. Totals data are summarised as percentages of available data. #: 58% of patients smoked more than 10 pack-years. The proportion of ever smoking or active smoking patients is unknown. ¶: 90% of patients smoked more than 10 pack-years. The proportion of ever smoking or active smoking patients is unknown.

      • TABLE 3

        Treatment strategies and outcomes

        Study (last name first author)TubbsCarringtonYousemAkiraTravisRyuBaloiraKawabataIndividual casesTotal
        Treatment–26/40 (65)–––21/23 (91)12/12 (100)31/31 (100)61/67 (91)151/173 (87)
         Removal of exposure–––––4/27 (15)17/31 (55)6/61 (10)27/119 (23)
         Prednisolone–––––21/23 (91)9/12 (75)31/31 (100)58/67 (86)119/133 (89)
         Other first/second line treatment––––––3/12 (25)–12/67 (18)15/79 (19)
        Follow-up years–3.1±2.89 (0.8–22)3.2±1.3–3.1±3.71.9 (0.8–5.8)8.25––
        Outcome
         Improvement7/14 (50)16/26 (62)14/25 (56)2/6 (33)–1/19 (5)4/8 (50)30/31 (97)+37/66 (57)111/195 (57)
         Stable–3/26 (11)3/25 (12)3/6 (50)12/18 (67)12/19 (63)2/8 (25)–5/66 (8)37/168 (22)
         Worsened7/14 (50)7/26 (27)8/25 (32)1/6 (147)5/18 (28)1/19 (5)2/8 (25)5/14 (36)–26/130 (20)
         Death––––1/18 (6)#5/19 (25)¶–1/31 (3)14/66 (22)21/134 (16)
         Transplantation––––––––2/66 (3)2/66 (3)
         Relapse––––––––12/62 (18)12/62 (18)
        PFT
         Normalised––––––––7/39 (18)7/39 (18)
         Improved––––––6/9 (67)–15/39 (38)16/48 (33)
         Stabilised––––––1/9 (11)–10/39 (26)11/48 (23)
         Deteriorated––––––2/9 (22)–7/39 (18)9/48 (19)

        Data are presented as n/n (%), mean, mean±sd, or mean (range). Available data from case series and individual cases are presented. Totals data are summarised as percentages of available data. PFT: pulmonary function test; CT: computed tomography. #: patient committed suicide; ¶: two patients died of cancer; +: five out of 14 patients with follow-up CT developed honeycombing during follow-up. Outcomes were not uniformly good as suggested by these data.

        Supplementary Materials

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          Please note: supplementary material is not edited by the Editorial Office, and is uploaded as it has been supplied by the author.

          Supplementary_Table_1.pdf ERR-0181-2019_Supplementary_Table_1

          Supplementary_Table_2.pdf ERR-0181-2019_Supplementary_Table_2

          Supplementary_Table_3.pdf ERR-0181-2019_Supplementary_Table_3

          Supplementary_Figure_1.pdf ERR-0181-2019_Supplementary_Figure_1

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        Desquamative interstitial pneumonia: a systematic review of its features and outcomes
        Merel E. Hellemons, Catharina C. Moor, Jan von der Thüsen, Mariska Rossius, Arlette Odink, Laila Haugen Thorgersen, Johny Verschakelen, Wim Wuyts, Marlies S. Wijsenbeek, Elisabeth Bendstrup
        European Respiratory Review Jun 2020, 29 (156) 190181; DOI: 10.1183/16000617.0181-2019

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        Desquamative interstitial pneumonia: a systematic review of its features and outcomes
        Merel E. Hellemons, Catharina C. Moor, Jan von der Thüsen, Mariska Rossius, Arlette Odink, Laila Haugen Thorgersen, Johny Verschakelen, Wim Wuyts, Marlies S. Wijsenbeek, Elisabeth Bendstrup
        European Respiratory Review Jun 2020, 29 (156) 190181; DOI: 10.1183/16000617.0181-2019
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