Bhargava Abhilasha, Khedkar Kiran
General Surgery, Jawaharlal Nehru Medical College, Datta Meghe Institute of Higher Education and Research, Wardha, IND.
Pediatric Surgery, All India Institute of Medical Sciences, Nagpur, IND.
Cureus. 2024 May 14;16(5):e60315. doi: 10.7759/cureus.60315. eCollection 2024 May.
Hirschsprung disease, a rare genetic disorder affecting the enteric nervous system, is characterized by the absence of ganglion cells in the myenteric plexus. Typically identified in neonates due to the failure to pass meconium, diagnosis beyond the first year of life is considered delayed. Common clinical manifestations in children with late-onset Hirschsprung disease include abdominal distension, abdominal pain, vomiting, fever, and abnormal bowel sounds. Sigmoid volvulus, though uncommon, can complicate Hirschsprung disease, potentially leading to misdiagnosis and severe complications such as intestinal perforation, hemorrhage, sepsis, and even mortality. Non-surgical interventions such as antibiotic therapy, intestinal decompression, and fluid resuscitation are preferred initial treatments to stabilize the patient. This case involves a 9-year-old boy who has presented with abdominal distension since birth and a lengthy history of irregular bowel habits. The diagnosis of Hirschsprung disease was confirmed at our institution, and the patient underwent a two-stage repair procedure, which was completed without any intraoperative or postoperative complications. The patient experienced an uneventful recovery, was discharged with stable vital signs, and regained normal bowel function. This case highlights the challenges of delayed diagnosis at nine years and underscores the importance of prompt management.
先天性巨结肠症是一种影响肠神经系统的罕见遗传性疾病,其特征是肌间神经丛中缺乏神经节细胞。通常在新生儿期因未能排出胎粪而被发现,一岁以后的诊断被认为是延迟诊断。迟发性先天性巨结肠症患儿的常见临床表现包括腹胀、腹痛、呕吐、发热和肠鸣音异常。乙状结肠扭转虽然不常见,但可使先天性巨结肠症复杂化,可能导致误诊以及肠穿孔、出血、败血症甚至死亡等严重并发症。抗生素治疗、肠道减压和液体复苏等非手术干预是稳定患者病情的首选初始治疗方法。该病例涉及一名9岁男孩,自出生以来一直腹胀,并有长期不规则排便习惯的病史。在我们机构确诊为先天性巨结肠症,患者接受了两阶段修复手术,手术完成后未出现任何术中或术后并发症。患者恢复顺利,生命体征平稳出院,肠道功能恢复正常。该病例凸显了9岁时延迟诊断的挑战,并强调了及时治疗的重要性。