Case Report: Sudden Fatal Hemorrhage in Ulcerative Fungal Laryngotracheitis—A Pediatric Case Report

In this report, we describe an autopsy case of a child affected by acute lymphoblastic leukemia and opportunistic pulmonary aspergillosis. The patient died because of a full-thickness tracheal wall ulceration with right inferior thyroid artery lesion and sudden hemorrhage, likely ascribable to undiagnosed invasive Aspergillus laryngotracheitis. Aspergillus infection, particularly in immunocompromised patients, should be considered an urgent risk factor to manage as it may lead to sudden fatal events in absence of evident critical symptoms.

Case report: fatal bleeding from a duodenal ulcer—Dieulafoy’s lesion?

A 46-year-old man was admitted to the hospital by ambulance due to syncope. A standard blood screening showed a normal Hb value. The man had known hemorrhoids and a single fresh rectal bleeding earlier at home. On the following morning, the patient suddenly required resuscitation within a few minutes and subsequently died. Autopsy revealed a fatal hemorrhage with blood loss in the stomach and small and large intestines and a mucosal defect of the duodenum. After autopsy, the question arose whether the cause of death might have been a rare Dieulafoy’s lesion—aim of this case report was to clarify the diagnosis.

Circulatory neutrophils exhibit enhanced neutrophil extracellular trap formation in early puerperium: NETs at the nexus of thrombosis and immunity?

Pregnancy is associated with elevated maternal levels of cell-free DNA of neutrophil extracellular trap (NET) origin, as circulatory neutrophils exhibit increased spontaneous NET formation in vitro, mainly driven by G-CSF and finely modulated by sex hormones. The postpartum period, on the other hand, involves physiological alterations consistent with the need for protection against infections and fatal hemorrhage. Our findings indicate that all relevant serum markers of neutrophil degranulation and NET release are substantially augmented postpartum. Neutrophil pro-NETotic activity in vitro is also upregulated particularly in post-delivery neutrophils. Moreover, maternal puerperal neutrophils exhibit a strong pro-NETotic phenotype, associated with increased levels of all key players in the generation of NETs - citH3, MPO, NE, and ROS, compared to non-pregnant and pregnant controls. Intriguingly, post-delivery NET formation is independent of G-CSF in contrast to late gestation and complemented by the presence of TF on the NETs, alterations in the platelet activation status, and activation of the coagulation cascade, triggered by circulating microparticles. Taken together, our results reveal the highly pro-NETotic and potentially procoagulant nature of postpartum neutrophils, bridging an overt immune activation with possible harmful thrombotic incidence.

Recurrent Artery of Heubner Aneurysm Masquerading as Caudate Hemorrhage without Subarachnoid Hemorrhage in Moyamoya Disease: A Case Report and Literature Review

Background: Clinically, the recurrent artery of Heubner (RAH) aneurysm is extremely rare, commonly presents with subarachnoid hemorrhage (SAH). Case Report: A 73-year-old man with a known moyamoya disease who presented as caudate hemorrhage attributable to an incidental flow aneurysm distal on the right RAH, which was managed conservatively after an unsuccessful endovascular attempt. Unfortunately, the patient died five weeks after hospital discharge because of re-rupture of the aneurysm. To the best of our knowledge, the RAH aneurysm manifesting as caudate hemorrhage without SAH has not previously been reported. Conclusion: This case highlights that the RAH aneurysm masquerading as caudate hemorrhage without SAH is exceedingly rare but can be encountered, representing a diagnostic and therapeutic challenge, and should be considered in the differential diagnosis. Moreover, early identifying and then eliminating such vascular anomaly if possible is of importance to prevent fatal hemorrhage.

Case Report: A Case of Infant Bronchial Dieulafoy's Disease and Article Review

Background: Bronchial Dieulafoy's disease (BDD), characterized by constant diameter arterial malformation, is rare, especially among infants. The pathogenesis and clinical features of pediatric patients are unknown. Misdiagnosis and biopsy operations may lead to potential massive hemorrhage, which endangers the patient's life.Case Presentation: Here, we present a case of a 9-month-old boy who was diagnosed with BDD with massive hemoptysis. The boy was cured by embolization of the bronchial artery and was in good health at the 1-year follow-up. In addition, we searched PubMed, Google Scholar, and Web of Science databases using keyword “Bronchial Dieulafoy's Disease (BDD)” and found six additional cases of pediatric BDD.Conclusion: It is still insufficient to draw a conclusion about the origin of the disease. Bronchial angiography and endobronchial ultrasonography are considered promising methods to diagnose Dieulafoy's disease of the bronchus. Bronchoscopy with transbronchial biopsy should not be deployed due to the high risk of fatal hemorrhage. Explicit clinical case reports of BDD are needed to enhance the understanding of this rare disease.

Fatal hemorrhage after tonsillectomy

This is the first report of a segmental mediolysis occurring in arterioles, affecting the neck/tonsillar region and leading to a fatal hemorrhage after a tonsillectomy in a 20-year-old woman. Arteriolar mediolysis could be observed in tissue layers about 2 cm distant from the tonsillectomy region. Reparative changes were observed in the carotid and small neck arteries as well as in only one intramyocardial arteriole; however, no other vascular regions typically reported in segmental mediolysis arteriopathy were affected and aneurysm formation was not observed. No family history of vascular disease or hypertension was known. Pathophysiologically, previously reported reasons for post-tonsillectomy bleeding could be excluded. Segmental mediolysis may represent a wider spectrum of affected arteriolar vessels besides the reported small and medium sized arteries leading to aneurysms, which impedes radiographic diagnosis.

Unsteady Hemodynamics in Intracranial Aneurysms With Varying Dome Orientations

Fluid loading within an intracranial aneurysm is difficult to measure but can be related to the shape of the flow passage. The outcome of excessive loading is a fatal hemorrhage, making it necessary for early diagnosis. However, arterial diseases are asymptomatic and clinical assessment is a challenge. A realistic approach to examining the severity of wall loading is from the morphology of the aneurysm itself. Accordingly, this study compares pulsatile flow (Reynolds number Re = 426, Womersley number Wo = 4.7) in three different intracranial aneurysm geometries. Specifically, the spatio-temporal movement of vortices is followed in high aspect ratio aneurysm models whose domes are inclined along with angles of 0, 45, and 90 deg relative to the plane of the parent artery. The study is based on finite volume simulation of unsteady three-dimensional flow while a limited set of particle image velocimetry experiments have been carried out. Within a pulsatile cycle, an increase in inclination (0–90 deg) is seen to shift the point of impingement from the distal end toward the aneurysmal apex. This change in flow pattern strengthens helicity, drifts vortex cores, enhances spatial displacement of the vortex, and generates skewed Dean's vortices on transverse planes. Patches of wall shear stress and wall pressure shift spatially from the distal end in models of low inclination (0–45 deg) and circumscribe the aneurysmal wall for an inclination angle of 90 deg. Accordingly, it is concluded that high angles of inclination increase rupture risks while lower inclinations are comparatively safe.

Comparative Study Between Haemorrhage and Surgical Emphysema as Complication of Emergency Tracheostomy

Background: Emergency tracheostomy is the commonest surgical intervention performed as lifesaving procedure by ENT surgeons on patients in emergency room of ENT and ICU. Several operative and postoperative complications have been reported after tracheostomy including fatal hemorrhage. Massive subcutaneous neck emphysema occurred because ventilation started at the time when the hemorrhage was not completely managed and the tracheal tube was not fully secured. Objective: This study was conducted to compare haemorrhage and surgical emphysema as complication of emergency tracheostomy among patients admitted in Department of ENT and Head-Neck Surgery in Dhaka Medical College & Hospital. Methods and Materials: This was a Cross Sectional Study conducted in the ENT and Head- Neck Surgery Department, Dhaka Medical College Hospital from July 2015 to June 2016. A total of 90 patients were included who fulfilled the selection criteria. Non probability convenient sampling was used to collect data. Statistical analysis was done by SPSS version 21. Results: Mean± SD of the study subjects was 49.84±17.4 years with most patients belonged to 40-79 years age group. 28.9% patients had habit of smoking and betel nut chewing. (48.9%) patients suffered from Ca larynx. Out of 90 tracheostomy patients, 40(44.4%) patients had had different complications including haemorrhage (13.3%), surgical emphysema (10%), haemorrhage with surgical emphysema (6.7%), tube blockage (3.3%), wound infection (3.3%) and injury to local tissue (2.2%). Haemorrhage occurs from anterior jugular vein, middle thyroid veins and thyroid gland and included peroperative haemorrhage (7.8%), immediate post- operative haemorrhage, <24 hours, (4.43%) and late post-operative haemorrhage (1.2%). Conclusion: In this study, haemorrhage followed by surgical emphysema and haemorrhage with surgical emphysema were found as major complications following emergency tracheostomy, J Dhaka Medical College, Vol. 28, No.2, October, 2019, Page 159-163

Apatinib in patients with recurrent or metastatic adenoid cystic carcinoma of the head and neck: a single-arm, phase II prospective study

Background: Apatinib, a vascular endothelial growth factor receptor (VEGFR) blocker, has demonstrated encouraging antitumor activities and tolerable toxicities in various cancer types. Recurrent or metastatic adenoid cystic carcinoma of the head and neck (R/MACCHN) carries a poor prognosis, and treatment options are currently limited. This study was conducted to explore the antitumor activity and safety of apatinib in patients with R/MACCHN. Methods: In this phase II single-arm, prospective study, patients aged 15–75 years with incurable R/MACCHN received apatinib at a 500 mg dose once daily until intolerance or progression occurred. The primary endpoint was the 6-month progression-free survival (PFS) rate based on RECIST version 1.1. The secondary endpoints included response rate, overall survival (OS), and safety. Efficacy was assessed in all dosed patients with at least one post-baseline tumor assessment. Results: Among 68 patients treated with apatinib, 65 were evaluable for efficacy analysis, with a median follow-up time of 25.8 months. The 6-month, 12-month, and 24-month PFS rates were 92.3% [95% confidence interval (CI): 83–97.5%], 75.2% (95% CI: 61.5–84.0%) and 44.7% (95% CI: 32.3–57.5%), respectively. The objective response rate (ORR) and disease control rate (DCR), as assessed by investigators, were 46.2% (95% CI: 33.7–59.0%) and 98.5% (95% CI: 91.7–100.0%), respectively. The median duration of response was 17.7 months [interquartile range (IQR) 14.0–20.9]. The 12-month and 24-month OS rates were 92.3% (95% CI: 83.0–97.5%) and 82.3% (95% CI: 70–90.4%), respectively. The most common adverse events of grades 3–4 were hypertension (5.9%), proteinuria (9.2%), and hemorrhage (5.9%). One patient developed a fatal hemorrhage. Conclusion: An encouraging PFS, a high ORR, and a manageable safety profile were observed in this study. It seems that the administration of apatinib in R/MACCHN is likely to have a clinically meaningful therapeutic benefit and warrants further investigation. This study was prospectively registered in ClinicalTrials.gov (NCT02775370; date of registration: 17 May 2016; date of first patient enrollment: 25 May 2016)