RESUMO
Solitary caecal diverticulae are rare in children and presentation with massive gastrointestinal (GI) bleeding is seldom reported. We present the case of a 13-year-old boy with a two-year history of abdominal pain and multiple inconclusive investigations presenting with a life threating lower GI bleed. We also review the literature surrounding solitary caecal diverticulae and caecal duplication cysts (CDCs).
Assuntos
Divertículo , Hemorragia Gastrointestinal , Dor Abdominal/diagnóstico , Dor Abdominal/etiologia , Adolescente , Criança , Divertículo/complicações , Divertículo/diagnóstico , Hemorragia Gastrointestinal/diagnóstico , Hemorragia Gastrointestinal/etiologia , Humanos , MasculinoRESUMO
OBJECTIVE: To evaluate the effect of combining antenatal sildenafil with fetal tracheal occlusion (TO) in fetal rabbits with surgically induced congenital diaphragmatic hernia (CDH). BACKGROUND: Although antenatal sildenafil administration rescues vascular abnormalities in lungs of fetal rabbits with CDH, it only partially improves airway morphometry. We hypothesized that we could additionally stimulate lung growth by combining this medical treatment with fetal TO. METHODS: CDH was created on gestational day (GD)23 (n=54). Does were randomized to receive either sildenafil 10âmg/kg/d or placebo by subcutaneous injection from GD24 to GD30. On GD28, fetuses were randomly assigned to TO or sham neck dissection. At term (GD30) fetuses were delivered, ventilated, and finally harvested for histological and molecular analyses. Unoperated littermates served as controls. RESULTS: The lung-to-body-weight ratio was significantly reduced in sham-CDH fetuses either (1.2â±â0.3% vs 2.3â±â0.3% in controls, P=0.0003). Sildenafil had no effect on this parameter, while CDH fetuses undergoing TO had a lung-to-body-weight ratio comparable to that of controls (2.5â±â0.8%, P<0.0001). Sildenafil alone induced an improvement in the mean terminal bronchiolar density (2.5â±â0.8âbr/mm2 vs 3.5â±â0.9âbr/mm2, P=0.043) and lung mechanics (static elastance 61â±â36âcmH2O /mL vs 113â±â40âcmH2O/mL, P=0.008), but both effects were more pronounced in fetuses undergoing additional TO (2.1â±â0.8âbr/mm2, P=0.001 and 31â±â9âcmH2O/mL, P<0.0001 respectively). Both CDH-sham and CDH-TO fetuses treated with placebo had an increased medial wall thickness of peripheral pulmonary vessels (41.9â±â2.9% and 41.8â±â3.2%, vs 24.0â±â2.9% in controls, P<0.0001). CDH fetuses treated with sildenafil, either with or without TO, had a medial thickness in the normal range (29.4%â±â2.6%). Finally, TO reduced gene expression of vascular endothelial growth factor and surfactant protein A and B, but this effect was counteracted by sildenafil. CONCLUSION: In the rabbit model for CDH, the combination of maternal sildenafil and TO has a complementary effect on vascular and parenchymal lung development.
Assuntos
Hérnias Diafragmáticas Congênitas , Pulmão/crescimento & desenvolvimento , Citrato de Sildenafila/administração & dosagem , Traqueia/cirurgia , Animais , Terapia Combinada , Modelos Animais de Doenças , Feminino , Feto , Gravidez , Coelhos , Distribuição AleatóriaRESUMO
Congenital diaphragmatic hernia is surgically correctable, yet the poor lung development determines mortality and morbidity. In isolated cases the outcome may be predicted prenatally by medical imaging. Cases with a poor prognosis could be treated before birth. However, prenatal modulation of lung development remains experimental. Fetoscopic endoluminal tracheal occlusion triggers lung growth and is currently being evaluated in a global clinical trial. Prenatal transplacental sildenafil administration may in due course be a therapeutic approach, reducing the occurrence of persistent pulmonary hypertension, either alone or in combination with fetal surgery.