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Management and Long-Term Outcomes of Persistent Tachypnea of Infancy/Neuroendocrine Cell Hyperplasia of Infancy: A
Honorata Marczak1, Katarzyna Krenke1, Matthias Griese2
1Department of Pediatric Pneumology and Allergy, Medical University of Warsaw, Warsaw, Poland.
Insights
Persistent tachypnea of infancy (PTI), or neuroendocrine cell hyperplasia of infancy (NEHI), management varies across Europe. While most children improve, some experience long-term symptoms, indicating a need for standardized guidelines.
Area of Science:
- Pediatric Pulmonology
- Childhood Interstitial Lung Diseases
Background:
- Persistent tachypnea of infancy (PTI), also known as neuroendocrine cell hyperplasia of infancy (NEHI), is a common childhood interstitial lung disease.
- Current management protocols lack standardization, and long-term outcome data are limited.
Purpose of the Study:
- To investigate treatment strategies for PTI/NEHI across European countries.
- To analyze variations in clinical management and long-term patient outcomes.
Main Methods:
- A European, multicenter, retrospective, observational study.
- Data on clinical characteristics, interventions, and follow-up were collected from 378 children across 17 countries.
- Treatment strategies were compared among countries with at least 10 patients.
Main Results:
- Common interventions included oxygen supplementation (75.9%), inhaled therapies (62.4%), systemic glucocorticoids (37.0%), and nutritional support (33.8%).
- Significant variability in treatment practices was observed across European countries (p<0.05).
- While most children improved by age 4, a subset experienced persistent hypoxemia, exercise intolerance, and abnormal lung function into adolescence.
Conclusions:
- Treatment strategies for PTI/NEHI differ significantly across European nations, underscoring the need for evidence-based guidelines.
- The long-term prognosis for PTI/NEHI is generally favorable, but ongoing monitoring is essential due to potential residual symptoms in some individuals.
Background:
Persistent tachypnea of infancy (PTI), also known as neuroendocrine cell hyperplasia of infancy (NEHI), represents 1 of the most common childhood interstitial lung diseases. Despite its frequency, standardized management protocol is lacking, and long-term outcome data remain limited.
Research Question:
What treatment is used for patients with PTI/NEHI, how does clinical management vary across European countries, and what are the long-term outcomes in affected patients?
Study Design And Methods:
This was a European multicenter, retrospective, observational study. Clinical characteristics, therapeutic interventions, and long-term follow-up data were collected and analyzed. Treatment strategies were compared among countries that contributed at least 10 patients.
Results:
A total of 378 children (63.5% male [240 of 378]) from 73 centers across 17 countries were enrolled, with a median age at diagnosis of 9 months (interquartile range [IQR], 6-13 months). Therapeutic interventions included oxygen supplementation (75.9% [287 of 378]); inhaled bronchodilators, inhaled glucocorticoids, or both (62.4% [236 of 378]); systemic glucocorticoids (37.0% [140 of 378]); and nutritional support (33.8% [128 of 378]). Of the children who received oxygen therapy, 53.6% (154 of 287) were reported to have been weaned off, with a median age at weaning of 24 months (IQR, 16-36 months). Marked variability in treatment practices was observed across participating countries (P < .05). Longitudinal data were available for 48.9% of patients (185 of 378) with a median follow-up of 19 months (IQR, 16-57 months). The proportion of symptomatic children declined over time, with the most marked improvement observed at 4 years of age. Resolution of imaging and pulmonary function abnormalities also was reported; however, a subset of patients continued to demonstrate persistent hypoxemia, crackles, and exercise intolerance, as well as abnormal imaging and pulmonary function test findings into adolescence.
Interpretation:
Our results show that significant differences in treatment strategies for PTI/NEHI were observed across European countries, highlighting the need for evidence-based guidelines. Although long-term prognosis generally is favorable, residual symptoms remain in some patients, warranting continued follow-up.
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