RESUMEN
This article reports a patient with intra-abdominal hemorrhage secondary to a rare vascular disease, segmental arterial mediolysis (SAM). The patient was a 68-year-old male who presented with chilling and severe abdominal pain. Abdominal computed tomography results suggested the presence of an intra-abdominal hemorrhage. Although visceral angiography illustrated multiple aneurysms in the branches of the hepatic artery, active bleeding was not evident. Conservative therapy including transfusion was performed, and re-angiography revealed the disappearance of multiple hepatic arterial aneurysms 8 months later. This is the first case of intra-abdominal hemorrhage related to SAM of the hepatic artery in which natural history of SAM was seen.
Asunto(s)
Arteria Hepática/patología , Túnica Media/patología , Enfermedades Vasculares/patología , Anciano , Humanos , Masculino , Rotura Espontánea , Tomografía Computarizada por Rayos XAsunto(s)
Apendicitis/etiología , Colon/anomalías , Quistes/complicaciones , Perforación Intestinal/etiología , Adolescente , Apendicectomía , Apendicitis/diagnóstico por imagen , Quistes/diagnóstico por imagen , Femenino , Humanos , Perforación Intestinal/cirugía , Tomografía Computarizada por Rayos XRESUMEN
BACKGROUND/AIMS: We report a case of solitary Peutz-Jeghers-type hamartomatous polyp of the duodenum in a 22-year-old Japanese woman along with the results of genomic analysis. METHODS/RESULTS: The patient was almost asymptomatic, though endoscopic examination revealed a solitary lobular polypoid lesion measuring 3 cm in diameter in the first portion of the duodenum. The lesion was resected endoscopically. Histopathological examination showed hyperplasia with a tree branch-like extension of the lamina propria derived from the muscularis mucosae, consistent with histological features of polyps of Peutz-Jeghers syndrome (PJS). No mucocutaneous pigmentation of the skin was evident and family history was negative. Analysis of the loss of heterozygosity at the locus of 19p 13.3 and mutation analysis of the STK11/LKB1 gene, which has recently been recognized as a susceptible gene in PJS, were performed. However, no evidence of genomic abnormality was found. CONCLUSION: The clinical and investigative findings in our case suggest that the solitary Peutz- Jeghers-type hamartomatous polyp can be regarded as a clinical entity separate from PJS.
