Idiopathic Pulmonary Hemorrhage in Infancy: A Case Report and Literature Review

Abstract

Acute idiopathic pulmonary hemorrhage in infants (AIPHI) is a rare and quite low-described entity. Nowadays, pathophysiological mechanisms are poorly understood, although the lethality remains high. We present an autopsy case report of a 2-day-old male who developed respiratory distress and blood leakage from the endotracheal tube (ET) and suddenly died because of acute pulmonary hemorrhage. A postmortem examination and histological analysis were performed and are reported in this paper. Alveolar spaces were filled with red blood cells and hyaline membranes in all the examined samples. The absence of other findings led us to select a post-mortem diagnosis of AIPHI. To support our diagnosis, we conducted a systematic review of the updated scientific literature and found that only 61 cases have been reported. Most of them presented acute respiratory distress and bleeding from the upper airways with blood leakage from ET (9.83%), hemoptysis (52.45%), epistaxis (8.2%), and hematemesis (3.27%). The autopsy data revealed hemorrhages of the lower airways and hemosiderin-laden macrophages. The data from the scientific publications and our findings are essential to achieving a correct diagnosis. On these bases, we suggest autoptic criteria to achieve a post-mortem diagnosis of AIPHI.

Sudden Death: A Practical Autopsy Approach to Unexplained Mediastinitis Due to Fatal Untreated Neck Infections—A Systematic Review

“…Dissections should be performed with extreme caution. The first step is to make the classic jugolo-pubic incision useful in both adult and child cases [ 34 ]. Putrefaction could alter the thickness and elasticity of the skin; therefore, we suggest carefully flipping the skin layer and the platysma muscle together in order to expose the underlying muscles.…”

Traumatic Aneurysm Involving the Posterior Communicating Artery

“…The rupture of a pre-existing aneurysm or a previously healthy vessel can be caused by aggression. In cases like this, macroscopic neuropathology examination is very important to ensure targeted microscopic findings [8][9][10], because it is difficult to assess whether the rupture of the aneurysm is due to a stress-related increase in blood pressure or by blunt trauma, or the consequence of a mixed mechanism [11]. Furthermore, assessing the cocaine and benzoylecgonine concentration can assist with the interpretation of intracranial hemorrhage in drug abusers [12].…”

Interstitial Lung Disease in Neonates: A Long Road Is Being Paved

“…The recently published remarkable study on acute idiopathic pulmonary hemorrhage in infants (AIPHI), has shed the lights on the idiopathic pattern of lung pathology, and suggested diagnostic criteria that consists of postmortem macroscopic and histological assessment for AIPHI, as clinical assessment is not enough to explain the pattern of the disease [ 6 ]. Tachypnea, cough, hypoxemia, crackles, failure to thrive, lung imaging irregularities, and impaired lung function, although they are not solely pathognomonic of pulmonary disease, they are among the common clinical findings that are seen in ILD in children [ 7 , 8 ].…”