Long-term outcomes of persistent tachypnoea of infancy/neuroendocrine cell hyperplasia of infancy: study of the CRC chILD-EU

<bold>Background:</bold> Long-term data on the clinical course of persistent tachypnea of infancy (PTI)/neuroendocrine cell hyperplasia of infancy (NEHI) remain limited. <bold>Aim:</bold> To evaluate long-term outcomes of PTI/NEHI patients. <bold>Methods:</bold> Among chILD-EU participating countries, clinical data of PTI/NEHI patients from diagnosis to age 13 were analyzed, including spirometry and chest CT results when available. The impact of systemic steroid (SS) treatment on symptoms at ages 2 and 4 was also assessed. <bold>Results:</bold> 185 patients (65% male, median diagnosis at 8 mo.) were included. Symptoms, chest CT, and spirometry abnormalities showed resolution over time (Table). Prevalence of tachypnea, retractions, hypoxemia, and crackles at ages 2 and 4 did not differ between SS-treated and untreated children. <bold>Conclusion:</bold> Most symptoms of PTI/NEHI resolve by age 4, although hypoxemia and crackles may persist into adolescence. Additional test abnormalities tend to normalize over time. SS treatment does not appear to affect symptom resolution. <table-wrap><object-id>erj;66/suppl_69/OA5423/TB1</object-id><object-id>T1</object-id><object-id>TB1</object-id><table><colgroup><col></col><col></col><col></col><col></col><col></col><col></col><col></col><col></col></colgroup><tbody><tr><td><bold>Visit</bold></td><td><bold>Baseline</bold></td><td><bold>18mo</bold></td><td><bold>2y</bold></td><td><bold>4y</bold></td><td><bold>6y</bold></td><td><bold>10y</bold></td><td><bold>13y</bold></td></tr><tr><td> <bold>Symptoms - n</bold> Tachypnoea - n (%) Retractions - n (%) Crackles - n (%) Hypoxemia - n (%) Failure to thrive - n (%) </td><td> <bold>185</bold> 180(97) 174(94) 157(85) 141(76) 83(45) </td><td> <bold>185</bold> 125(68) 101(55) 107(58) 113(61) 57(31) </td><td> <bold>153</bold> 62(41) 50(33) 72(47) 64(42) 33(22) </td><td> <bold>85</bold> 12(14) 8(9) 35(41) 25(29) 8(9) </td><td> <bold>52</bold> 7(13) 3(6) 19(37) 15(29) 4(8) </td><td> <bold>15</bold> 2(13) 0 5(33) 4(27) 4(27) </td><td> <bold>8</bold> 0 0 2(25) 1(13) 0 </td></tr><tr><td> <bold>Follow-up chest CT - n</bold> Similar - n (%) Partial resolution - n (%) Complete resolution - n (%) Progression - n (%) </td><td></td><td> <bold>11</bold> 4(36) 7(64) 0 0 </td><td> <bold>12</bold> 3(25) 6(50) 1(8) 2(17) </td><td> <bold>8</bold> 0 4(50) 4(50) 0 </td><td> <bold>10</bold> 2(20) 4(40) 4(40) 0 </td><td> <bold>4</bold> 0 3(75) 1(25) 0 </td><td> <bold>1</bold> 1(100) 0 0 0 </td></tr><tr><td> <bold>Spirometry - n</bold> Normal - n (%) ↓FVC - n (%) ↓FVC/FEV1 - n (%) </td><td></td><td></td><td></td><td> <bold>23</bold> 15(65) 8(35) 0 </td><td> <bold>39</bold> 27(69) 10(26) 2(5) </td><td> <bold>15</bold> 14(93) 1(7) 0 </td><td> <bold>8</bold> 8(100) 0 0 </td></tr></tbody></table></table-wrap>

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Published
2025-09-27
DOI
https://doi.org/10.1183/13993003.congress-2025.oa5423
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Neonatal Respiratory Health Research
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article

Long-term outcomes of persistent tachypnoea of infancy/neuroendocrine cell hyperplasia of infancy: study of the CRC chILD-EU

Maynor Bravo-López, Honorata Marczak, Joanna Peradzyńska, Sarah Mayell et al.
Neonatal Respiratory Health Research
article

Long-term outcomes of persistent tachypnoea of infancy/neuroendocrine cell hyperplasia of infancy: study of the CRC chILD-EU

Maynor Bravo-López, Honorata Marczak, Joanna Peradzyńska, Sarah Mayell, Winfried Baden, Philipp Latzin, Florian Samuel Halbeisen, Julia Carlens, Nicola Ullmann, Laura Petrarca, Elpis Hatziagorou, Silvia Castillo, Dafni Moriki, Kim G. Nielsen, Elias Seidl, Nadia Nathan, Juan A. López‐Andreu, Katarzyna Krenke, Christiane Lex, Lutz Naehrlich, Steve Cunningham, Marijke Proesmans, Joanna Lange, Dóra Krikovszky, Mirella Gáboli, Matthias Griese, Václav Koucký, Nazan Çobanoğlu, Brigitte W.M. Willemse, Nicolas Regamey, Nicolaus Schwerk, Anna Wiemers, Jana Vienna Rödler, Sevgi Pekcan, Anna Zschocke, Alba Torrent, Nuria Díez-Monge
article en

Abstract

<bold>Background:</bold> Long-term data on the clinical course of persistent tachypnea of infancy (PTI)/neuroendocrine cell hyperplasia of infancy (NEHI) remain limited. <bold>Aim:</bold> To evaluate long-term outcomes of PTI/NEHI patients. <bold>Methods:</bold> Among chILD-EU participating countries, clinical data of PTI/NEHI patients from diagnosis to age 13 were analyzed, including spirometry and chest CT results when available. The impact of systemic steroid (SS) treatment on symptoms at ages 2 and 4 was also assessed. <bold>Results:</bold> 185 patients (65% male, median diagnosis at 8 mo.) were included. Symptoms, chest CT, and spirometry abnormalities showed resolution over time (Table). Prevalence of tachypnea, retractions, hypoxemia, and crackles at ages 2 and 4 did not differ between SS-treated and untreated children. <bold>Conclusion:</bold> Most symptoms of PTI/NEHI resolve by age 4, although hypoxemia and crackles may persist into adolescence. Additional test abnormalities tend to normalize over time. SS treatment does not appear to affect symptom resolution. <table-wrap><object-id>erj;66/suppl_69/OA5423/TB1</object-id><object-id>T1</object-id><object-id>TB1</object-id><table><colgroup><col></col><col></col><col></col><col></col><col></col><col></col><col></col><col></col></colgroup><tbody><tr><td><bold>Visit</bold></td><td><bold>Baseline</bold></td><td><bold>18mo</bold></td><td><bold>2y</bold></td><td><bold>4y</bold></td><td><bold>6y</bold></td><td><bold>10y</bold></td><td><bold>13y</bold></td></tr><tr><td> <bold>Symptoms - n</bold> Tachypnoea - n (%) Retractions - n (%) Crackles - n (%) Hypoxemia - n (%) Failure to thrive - n (%) </td><td> <bold>185</bold> 180(97) 174(94) 157(85) 141(76) 83(45) </td><td> <bold>185</bold> 125(68) 101(55) 107(58) 113(61) 57(31) </td><td> <bold>153</bold> 62(41) 50(33) 72(47) 64(42) 33(22) </td><td> <bold>85</bold> 12(14) 8(9) 35(41) 25(29) 8(9) </td><td> <bold>52</bold> 7(13) 3(6) 19(37) 15(29) 4(8) </td><td> <bold>15</bold> 2(13) 0 5(33) 4(27) 4(27) </td><td> <bold>8</bold> 0 0 2(25) 1(13) 0 </td></tr><tr><td> <bold>Follow-up chest CT - n</bold> Similar - n (%) Partial resolution - n (%) Complete resolution - n (%) Progression - n (%) </td><td></td><td> <bold>11</bold> 4(36) 7(64) 0 0 </td><td> <bold>12</bold> 3(25) 6(50) 1(8) 2(17) </td><td> <bold>8</bold> 0 4(50) 4(50) 0 </td><td> <bold>10</bold> 2(20) 4(40) 4(40) 0 </td><td> <bold>4</bold> 0 3(75) 1(25) 0 </td><td> <bold>1</bold> 1(100) 0 0 0 </td></tr><tr><td> <bold>Spirometry - n</bold> Normal - n (%) ↓FVC - n (%) ↓FVC/FEV1 - n (%) </td><td></td><td></td><td></td><td> <bold>23</bold> 15(65) 8(35) 0 </td><td> <bold>39</bold> 27(69) 10(26) 2(5) </td><td> <bold>15</bold> 14(93) 1(7) 0 </td><td> <bold>8</bold> 8(100) 0 0 </td></tr></tbody></table></table-wrap>

Semmelweis University (HU), University of Bern (CH), University Medical Center Groningen (NL), Innsbruck Medical University (AT), University of Liverpool (GB), Ankara University (TR), Justus-Liebig-Universität Gießen (DE), National and Kapodistrian University of Athens (GR), Charles University (CZ), Aristotle University of Thessaloniki (GR), Medical University of Warsaw (PL), University Hospital of Bern (CH), Copenhagen University Hospital (DK), Hospital Universitari i Politècnic La Fe (ES), Medizinische Hochschule Hannover (DE), Sorbonne Université (FR), Management Center Innsbruck (AT), Hôpital Armand-Trousseau (FR), Maternal and Child Health Hospital of Xinjiang Uygur Autonomous Region (CN), University Hospital in Motol (CZ), Vall d'Hebron Institut de Recerca (ES), Necmettin Erbakan University (TR), Ippokrateio General Hospital of Thessaloniki (GR), Universitätsmedizin Göttingen (DE), Bambino Gesù Children's Hospital (IT), University Children's Hospital Tübingen (DE), University Children's Hospital Zurich (CH), Instituto de Biomedicina de Sevilla (ES), St. Josef-Hospital (DE), Centre for Inflammation Research (GB), Alder Hey Children's Hospital (GB), INCLIVA Health Research Institute (ES), Hippocration General Hospital (GR), University of Duisburg-Essen (DE), Ludwig-Maximilians-Universität München (DE), KU Leuven (BE)
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