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• W2415996212 abstract "We read with interest the article titled “Update on Diffuse Lung Disease in Children” from Vece and Young1Vece T.J. Young L.R. Update on diffuse lung disease in children.Chest. 2016; 149: 836-845Abstract Full Text Full Text PDF PubMed Scopus (24) Google Scholar in a recent issue of CHEST (March 2016). We particularly appreciated their clear approach and recommendations about this heterogeneous and complex group of rare conditions. The exhaustive description of diagnosis, evaluations, and treatments is helpful, as most institutions will only manage a few cases per year and can be confused when faced with childhood interstitial lung disease (chILD). We would, however, like to raise concerns regarding the accuracy of CT scanning for the diagnosis of neuroendocrine cell hyperplasia of infancy (NEHI). This topic, which has also been mentioned in the European recommendations,2Bush A. Cunningham S. de Blic J. et al.chILD-EU CollaborationEuropean protocols for the diagnosis and initial treatment of interstitial lung disease in children.Thorax. 2015; 70: 1078-1084Crossref PubMed Scopus (157) Google Scholar mainly comes from the article of Brody et al,3Brody A.S. Guillerman R.P. Hay T.C. et al.Neuroendocrine cell hyperplasia of infancy: diagnosis with high-resolution CT.AJR Am J Roentgenol. 2010; 194: 238-244Crossref PubMed Scopus (108) Google Scholar in which images from 23 children with biopsy-proven NEHI were compared with six children with other chILD. In our cohort, we have had examples which contradict the fact that the CT scan pattern of “ground glass opacities in the right middle lobe, lingula, and medial segments of the upper and lower lobes, in the absence of other significant parenchymal abnormalities” is pathognomonic for NEHI.4Nathan N. Taam R.A. Epaud R. et al.A national internet-linked based database for pediatric interstitial lung diseases: the French network.Orphanet J Rare Dis. 2012; 7: 40Crossref PubMed Scopus (64) Google Scholar We have managed some patients with clinical chILD and with the same CT scan pattern (Fig 1) as described in NEHI. One case was a 2-year-old girl; another case involved a 6-month-old boy. They also presented with air-trapping, which is another sign of NEHI, both on CT scan and in pulmonary function test results. Both patients improved over years, as expected in NEHI5Deterding R.R. Pye C. Fan L.L. Langston C. Persistent tachypnea of infancy is associated with neuroendocrine cell hyperplasia.Pediatr Pulmonol. 2005; 40: 157-165Crossref PubMed Scopus (164) Google Scholar; however, they required corticosteroid therapy, which is unusual in NEHI. In addition, lung biopsy specimens from both patients at diagnosis exhibited a pattern of desquamative interstitial pneumonia with no bombesin-positive pulmonary neuroendocrine cells; this finding is contrary to the diagnosis of NEHI.6Rice A. Tran-Dang M.A. Bush A. Nicholson A.G. Diffuse lung disease in infancy and childhood: expanding the chILD classification.Histopathology. 2013; 63: 743-755Crossref PubMed Scopus (54) Google Scholar The patients were both subjected to sequencing of SFTPC, SFTPB, and ABCA3 proteins of surfactant. No genetic mutation was found in the 2-year-old girl, and a single mutation in the ABCA3 protein was found in the 6-month-old boy. Correct diagnosis of NEHI is of critical importance because, unlike other types of chILD, NEHI exhibits a poor response to corticosteroid therapy, requires other treatments, and has a better prognosis. On the basis of the two aforementioned examples, we think that the characteristic CT scan pattern of NEHI is not to be considered as pathognomonic. Protein surfactant mutations must be searched in all cases, and lung biopsies should still be considered for differential diagnosis. Update on Diffuse Lung Disease in ChildrenCHESTVol. 149Issue 3PreviewDiffuse lung diseases in children, also called children’s interstitial lung disease, are a diverse group of rare disorders that cause disturbances of gas exchange in the lungs. Although individually rare, there are many different forms of diffuse lung disease in children, and collectively these disorders are associated with significant morbidity and mortality, as well as health-care resource utilization. Over the past several years, there have been many significant advances in the field, including genetic discoveries and the development of clinical practice guidelines. Full-Text PDF ResponseCHESTVol. 149Issue 6PreviewWe thank Dr Berteloot and colleagues for their interest in our review article recently published in CHEST.1 The authors present two interesting cases as a platform to discuss a 2010 report of chest CT scan findings in neuroendocrine cell hyperplasia of infancy (NEHI).2 Berteloot et al raise concerns about the specificity of chest CT scans by proposing that their cases had imaging findings suggestive of NEHI but did not ultimately have NEHI based on the histologic findings and the clinical response to corticosteroid therapy. Full-Text PDF" @default.
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• W2415996212 title "Anything that Looks Like a Neuroendocrine Cell Hyperplasia of Infancy Is Not Necessarily a Neuroendocrine Cell Hyperplasia of Infancy" @default.
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