Clinical, functional, and computed tomography findings in a cohort of patients with neuroendocrine cell hyperplasia of infancy

Balinotti, J.; Maffey, Alberto; Colom, Alejandro; Roldán, Oscar; Díaz, Walter; Medín, Martín; Racimo, Mariano; Teper, Alejandro

doi:10.1002/ppul.25319

Cited by 12 publications

(16 citation statements)

References 26 publications

(81 reference statements)

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“…Our patient presented with the classic non-specific symptoms of NEHI described in prior case reports ( 1 – 8 ) including tachypnea, hypoxemia, retractions, and crackles on exam with poor weight gain related to increased caloric demands. Working diagnoses included prolonged and recurrent viral bronchiolitis as well as pneumonia, reactive airway disease, and obstructive sleep apnea.…”

Section: Discussionmentioning

confidence: 58%

“…Fortunately, prospective studies on children with NEHI demonstrate gradual improvement in symptoms over time, with associated decreased oxygen requirements ( 1 , 8 , 13 , 25 , 26 ). This was observed in our patient over subsequent months, with resultant weaning of bronchodilators and steroids and only 0.5 L of oxygen required at nighttime.…”

Section: Discussionmentioning

confidence: 99%

“…The accurate characterization of NEHI is critical given its implications for patient management and prognosis. The treatment for NEHI is primarily supportive, involving oxygen therapy and adjunctive therapies for associated comorbidities, which include gastroesophageal reflux, aspiration, poor growth, developmental delay, immune system abnormalities, and sleep apnea ( 2 , 3 , 8 , 11 , 21 , 22 ). Previous case reports have documented repeated hospitalizations, numerous comorbidities, and unnecessary treatments due to delayed diagnoses of NEHI given its similarity in presentation to other respiratory illnesses in infancy ( 2 – 4 , 6 , 7 ).…”

Section: Introductionmentioning

confidence: 99%

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Case report: Rare lung disease of infancy diagnosed with the assistance of a home pulse oximetry baby monitor

et al. 2022

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Neuroendocrine cell hyperplasia of infancy (NEHI) is a rare childhood interstitial lung disease characterized by a gradual onset of tachypnea, hypoxemia, and failure to thrive in the first 2 years of life. NEHI is challenging to diagnose and can masquerade as common respiratory infections and reactive airway disease. Timely diagnosis is essential to optimize management of comorbidities, improve outcomes, and prevent unnecessary interventions. We report a case of a 14-month-old male who was hospitalized multiple times with recurrent episodes of presumed bronchiolitis. However, early on, the parents had detected unexplained nighttime hypoxemia with a wearable home pulse oximetry baby monitor. While recurrent respiratory infections are common in infancy, our patient had numerous persistent symptoms refractory to traditional treatments, which prompted further workup and ultimately led to the diagnosis of NEHI. The home baby monitor provided useful information that accelerated workup for a presentation that did not fit the usual picture of recurrent bronchiolitis, bronchospasm, or pneumonia. These devices that monitor infant cardiopulmonary status and oxygenation are becoming increasingly popular for home use. There is controversy over their clinical utility due to the frequency of false alarms, excessive parental reliance on these devices, and lack of Food and Drug Administration oversight to ensure accuracy and effectiveness of these devices. Our case provides an example of how in certain clinical settings, information from these devices might serve as a complementary tool in the pediatrician’s medical decision-making and possibly lead to a rare diagnosis such as NEHI.

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Section: Discussionmentioning

confidence: 58%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Case report: Rare lung disease of infancy diagnosed with the assistance of a home pulse oximetry baby monitor

et al. 2022

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“…ere is no specific treatment for NEHI, but most of the patients require prolonged supplemental oxygen through the second year of life [14,19]. In our case, the patient currently is 34-month-old and continues to require supplemental oxygen at night.…”

Section: Discussionmentioning

confidence: 76%

“…In our case, the presence of prematurity was a confounder. NEHI in the setting of prematurity is very rare in the literature [14,19].…”

Section: Discussionmentioning

confidence: 99%

A Rare Case Report of NEHI in a Preterm Infant with Review of the Literature

Mangat

DeCoster

Milosavljevic

et al. 2022

Case Reports in Pediatrics

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Background. Neuroendocrine cell hyperplasia of infancy (NEHI) is a rare respiratory disorder. During infancy, it typically presents with hypoxemia, tachypnea, and respiratory distress, and is commonly misdiagnosed as common childhood illnesses such as pneumonia, reactive airway disease, or bronchiolitis. Lack of awareness about this relatively new and rare disorder in primary care and acute care settings lead to delayed diagnosis and unnecessary use of antibiotics. Case Presentation. We present a case of a 7-month-old girl, born prematurely at 32 weeks with tachypnea and respiratory distress who was initially diagnosed with viral pneumonia, then upper respiratory infection, and finally with community-acquired bacterial pneumonia, while the child never had any fever or upper respiratory symptoms. Failure of outpatient treatment with oral antibiotic and bronchodilator, with the persistence of respiratory symptoms such as retractions, bilateral crackles, and hypoxemia led to hospitalization for intravenous antibiotics. Given persistent symptoms, further evaluation was performed, and she was diagnosed with NEHI based on characteristic chest CT findings. Conclusions. Viral respiratory infections are the most frequent cause of respiratory illnesses in the first years of life. Primary care providers should be aware of less frequent causes of persistent respiratory symptoms in infancy like NEHI and other interstitial lung diseases in children. This may prevent unnecessary use of antibiotics and delayed diagnosis.

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Pulmonary function in children with persistent tachypnea of infancy

Marczak

Peradzyńska

Lange

et al. 2022

Pediatric Pulmonology

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Background: Data on the prevalence and type of lung function impairment in preschool and school-aged children previously diagnosed with persistent tachypnea of infancy (PTI) are scarce. Therefore, this study aims to assess pulmonary function in this age group. Methods: Children diagnosed with PTI over 3 years old were admitted for follow-up visits and healthy controls were enrolled. The study group included children who were able to complete pulmonary function tests (PFTs). Medical history, physical examination, and pulmonary function (spirometry, body plethysmography, impulse oscillometry, nitrogen multiple breath washout test, diffusing capacity for carbon monoxide [DL CO ]) were assessed.Results: Thirty-seven children (26 boys, 11 girls; median age: 5.6 years) diagnosed with PTI and 37 healthy controls were recruited. Forced expiratory volume in 1 s and forced vital capacity were significantly lower (−1.12 vs. 0.48, p = 0.002 and −0.83 vs. 0.31, p = 0.009, respectively); respiratory resistance at 5 Hz (0.06 vs. −0.62, p = 0.003), resonant frequency (1.86 vs. 1.36, p = 0.04), residual volume (RV) (2.34 vs. −1.2, p < 0.0001), RV%TLC (total lung capacity) (2.63 vs. −0.72, p < 0.0001), and specific airway resistance (5.4 vs. 2.59, p = 0.04) were significantly higher in PTI patients as compared with controls (data were presented as median z-score). Air trapping was found in 60.0%, and abnormally high lung clearance index and DL CO were found in 73.3% and 90.9% of PTI patients, respectively.Conclusions: This study demonstrated that lung function is affected in most children with PTI. PFTs showed that peripheral airways are the major zone of functional impairment.

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Clinical, functional, and computed tomography findings in a cohort of patients with neuroendocrine cell hyperplasia of infancy

Cited by 12 publications

References 26 publications

Case report: Rare lung disease of infancy diagnosed with the assistance of a home pulse oximetry baby monitor

Case report: Rare lung disease of infancy diagnosed with the assistance of a home pulse oximetry baby monitor

A Rare Case Report of NEHI in a Preterm Infant with Review of the Literature

Pulmonary function in children with persistent tachypnea of infancy

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