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Pediatr Pulmonol. 2019 Jun;54(6):822-827. doi: 10.1002/ppul.24301. Epub 2019 Mar 6.

Ground-glass burden as a biomarker in neuroendocrine cell hyperplasia of infancy.

Spielberg DR1,2,3, Brody AS4,5, Baker ML6,7, Woods JC1,2,4,8, Towe CT1,8.

Author information

1
Division of Pulmonary Medicine, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
2
Center for Pulmonary Imaging Research, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
3
Department of Pediatrics, Section of Pulmonary Medicine, Baylor College of Medicine, Houston, Texas.
4
Department of Radiology, University of Cincinnati College of Medicine, Cincinnati, Ohio.
5
Department of Radiology and Medical Imaging, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
6
Department of Pathology, Dartmouth-Hitchcock Medical Center, Lebanon, New Hampshire.
7
Department of Pathology and Laboratory Medicine, Geisel School of Medicine at Dartmouth, Hanover, New Hampshire.
8
Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio.

Abstract

BACKGROUND:

Neuroendocrine cell hyperplasia of infancy (NEHI) is a rare pediatric interstitial lung disease (ILD). Distinct chest computed tomography (CT) define its radiographic appearance-specifically, ground-glass (GG) opacities most prominent in the right middle lobe (RML) and lingula. We sought to quantitatively validate this description and correlate radiologic findings with clinical presentation.

METHODS:

Twenty-one children with NEHI were identified retrospectively, alongside 10 age-matched controls without lung disease. Clinical histories were reviewed for NEHI subjects. Semiautomated image analysis was used to measure lung volume and density. A patient-specific Hounsfield unit threshold defining GG was developed to quantify GG and assess its distribution in each subject.

RESULTS:

NEHI subjects had more GG than controls (37.9 ± 11.3% vs 14.0 ± 2.7%, P < 0.0001). The proportion of GG in the RML and lingula was greater in NEHI patients compared to controls (1.43 ± 0.37 vs 0.45 ± 0.21, P < 0.0001). GG preferentially involved the RML and lingula in 20/21 NEHI subjects. There was more GG distribution in NEHI subjects who were prescribed continuous oxygen compared with those using only nocturnal oxygen (45.7 ± 8.9% vs 29.3 ± 6.1%, P = 0.003).

CONCLUSIONS:

We confirm the previously reported finding that most patients with childhood ILD and a distinctive pattern of GG distribution on CT scan are likely to have NEHI. The amount of GG may be a biomarker for severity of respiratory disease.

KEYWORDS:

childhood interstitial lung disease; quantitative computed tomography

PMID:
30843378
DOI:
10.1002/ppul.24301

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