Evidence map›Paper›PMID 35678871›Full record

ArticleEuropean journal of pediatrics2022

French national cohort of neuroendocrine cell hyperplasia of infancy (FRENCHI) study: diagnosis and initial management.

Candice Fabre, Caroline Thumerelle, Morgane Dervaux, Rola Abou-Taam, Tiphaine Bihouee, Jacques Brouard, Annick Clement, Christophe Delacourt, Céline Delestrain, Ralph Epaud and 12 more

Abstract read
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In one paragraph

Article in European journal of pediatrics, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed, 1 pooled it
1.4field-weighted citation impact, top 19% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

4 citing papers in PubMed, 1 synthesis or guideline pooled it, 11 citations in OpenAlex.

  1. Old and emerging therapies for childhood interstitial lung disease (chILD): a systematic review.European respiratory review : an official journal of the European Respiratory Society · 2026
    Pooled it
  2. Article
  3. Interstitial lung disease in the newborn.Journal of perinatology : official journal of the California Perinatal Association · 2025
    Review
  4. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

22 authors at 13 institutions in 1 country.

Candice FabrePediatric Pulmonology Department, University Timone Hospital for Children, AP-HM, Marseille, France.
Caroline ThumerellePediatric Pulmonology and Allergy Department, Jeanne de Flandre Hospital, CHU Lille, Univ. Lille, Pole Enfant, Lille, France.
Morgane DervauxPediatric Pulmonology Department, University Timone Hospital for Children, AP-HM, Marseille, France.
Rola Abou-TaamAP-HP, Pediatric Pulmonology Department, Reference Center for Rare Lung Diseases, University Hospital of Necker-Enfants Malades, Paris, France.
Tiphaine BihoueeChronic Childhood Diseases Unit, Pediatric Department, Nantes University Hospital, Nantes, France.
Jacques BrouardService de Pédiatrie Médicale, CHU Caen, Caen, et UMR1311 DYNAMICURE, Normandie Université, UNICAEN, UNIROUEN, Rouen, France.
Annick ClementAP-HP, Pediatric Pulmonology Department and Reference Center for Rare Lung Diseases (RespiRare), Sorbonne University, Inserm UMR S-933 Childhood Genetic Disorders, Armand Trousseau Hospital, Paris, France.
Christophe DelacourtAP-HP, Pediatric Pulmonology Department, Reference Center for Rare Lung Diseases, University Hospital of Necker-Enfants Malades, Paris, France.
Céline DelestrainCenter for Rare Lung Diseases (RespiRare), Centre Hospitalier Intercommunal of Créteil, University Paris Est Créteil, INSERM, IMRB, Créteil, France.
Ralph EpaudCenter for Rare Lung Diseases (RespiRare), Centre Hospitalier Intercommunal of Créteil, University Paris Est Créteil, INSERM, IMRB, Créteil, France.
Sofiane GhdifanPediatric Unit, Grenoble-Alpes University Hospital, Grenoble, France.
Alice HadchouelAP-HP, Pediatric Pulmonology Department, Reference Center for Rare Lung Diseases, University Hospital of Necker-Enfants Malades, Paris, France.
Véronique HoudouinAP-HP, Pediatric Pulmonology Department, UMR S 976 INSERM Human Immunology, Paris University, Robert Debre Hospital, Paris, France.
Géraldine LabouretPediatric Pulmonology Department, University Hospital for Children, Toulouse, France.
Caroline PerissonPediatric Unit, University Hospital Sud Reunion, Saint-Pierre, France.
Philippe ReixPediatric Pulmonology, Allergology, Cystic Fibrosis Department, Hospices Civils of Lyon, Hôpital Femme Mère Enfant, Bron, France.
Marie-Catherine RenouxPaediatric Cardiology and Pulmonology Department, Montpellier University Hospital, Montpellier, France.
Françoise TroussierPediatric Department, University Hospital, Angers, France.
Laurence WeissSpecialized Pediatric Department, University Hospital of Strasbourg, Strasbourg, France.
Julie MazenqPediatric Pulmonology Department, University Timone Hospital for Children, AP-HM, Marseille, France.
Nadia NathanAP-HP, Pediatric Pulmonology Department and Reference Center for Rare Lung Diseases (RespiRare), Sorbonne University, Inserm UMR S-933 Childhood Genetic Disorders, Armand Trousseau Hospital, Paris, France.
Jean-Christophe DubusPediatric Pulmonology Department, University Timone Hospital for Children, AP-HM, Marseille, France. jean-christophe.dubus@ap-hm.fr.ORCID http://orcid.org/0000-0003-2078-4140
Inserm · FRAssistance Publique Hôpitaux de Marseille · FRAssistance Publique – Hôpitaux de Paris · FRAix-Marseille Université · FRCentre Hospitalier Universitaire de La Réunion · FRNormandie Université · FRUniversité Claude Bernard Lyon 1 · FRUniversité d'Angers · FRUniversité de Lille · FRUniversité de Montpellier · FRUniversité de Strasbourg · FRUniversité Fédérale de Toulouse Midi-Pyrénées · FRUniversité Grenoble Alpes · FR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Early diagnosis of neuroendocrine cell hyperplasia of infancy (NEHI) is crucial as, conversely to the other causes of intersititial lung disease, corticosteroids are not recommended. Diagnosis is historically based on lung biopsy (NEHI), but in current practice, a clinical and radiological approach is more and more preferred (NEHI syndrome). This national study aimed to address diagnosis and initial management of patients followed up for a NEHI pattern in pediatric centers for rare lung diseases (RespiRare, France). Data on neonatal and familial events, symptoms at diagnosis, explorations performed and results, and therapeutic management were collected by questionnaire. Fifty-four children were included (boys 63%). The mean onset of symptoms was 3.8 ± 2.6 months. The most frequent symptoms at diagnosis were tachypnea (100%), retraction (79.6%), crackles (66.7%), and hypoxemia (59.3%). The mean NEHI clinical score, evocative when ≥ 7/10, was 7.9 ± 1.4 (76% with a score ≥ 7). All chest CT-scans showed ground glass opacities evolving at least the middle lobe and the lingula. Lung biopsy was performed in 38.9% of the cases and was typical of NEHI in only 52.4%, even when the clinical presentation was typical. Initial treatments were oxygen (83.6%) and more curiously intravenous pulses of steroids (83.3%) and azithromycin (70.2%).

conclusionThis national cohort of patients underlines diagnosis difficulties of NEHI. A composite clinical and radiological score should help clinicians for limiting the use of anti-inflammatory drugs. WHAT IS KNOWN: •Neuroendocrine cell hyperplasia of infancy (NEHI) is an interstitial lung disease whose diagnosis is essential to limit corticosteroids therapy. WHAT IS NEW: •In this national cohort of 54 patients with a NEHI pattern, diagnosis is mainly based on clinical symptoms and chest CT-scan results. The newly proposed clinical score and, when performed, the lung biopsies are faulted in 25 and 50% of the cases, respectively. •Corticosteroids are widely used. Such results plead for a new composite score to formally diagnose NEHI.

Indexed as

Lung Diseases, InterstitialNeuroendocrine CellsChildHumansHyperplasiaInfantInfant, NewbornLungMaleRare DiseasesRetrospective StudiesAnti-inflammatory drugsChildhood interstitial lung diseaseChildrenClinical scoreCohortNeuroendocrine cell hyperplasia of infancy

Identifiers

PMID35678871
OpenAlexW4282926884

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.