A Case of Cecal Volvulus Presenting with Chronic Constipation in Lissencephaly
A 19-year-old male (height, 145 cm; weight, 34 kg), who was treated for cerebral palsy and refractory epilepsy due to lissencephaly, visited our hospital (Yeungnam University Medical Center, Daegu, Korea) because of severe abdominal distension and vomiting. Also, this patient was not able to perform independent daily living without any assistance because of mental disability. The patient had been treated with an anti-epileptic agent such as lamotrigine, phenobarbital, leviteracetam, rivotril, and ativan for epilepsy since he was a neonate and had visited our hospital several times for rectal decompression because of chronic constipation. The patient, suffered from chronic constipation over 3 years, had history of ≤2 bowel movements per week with hard large stool, necessitating rectal stimulation for defecation. Two months prior to admission, the patient had experienced a 3.0 kg weight loss and sleep disturbance with irritability.\nUpon admission, the patient was lethargic with dried lips. Abdominal examination showed epigastric and lower abdominal distension with decreased bowel sounds, which had mild rigidity. We could not check the site of pain and tenderness due to impaired communication with the patient. Laboratory findings showed leukocytosis with a white blood cell count of 45,690/µL, increased C-reactive protein at 15.14 mg/dL (reference range <0.5 mg/dL), and increased blood urea nitrogen/creatinine at 33.15/1.91 mg/dL. The patient had an electrolyte imbalance consisting of hyponatremia (124 mmol/L Na+) and hyperkalemia (5.9 mmol/L K+). Intravenous antibiotics of 3rd cephalosporin (50 mg/kg/day) and metronidazole (15 mg/kg/day) were administrated.\nAn initial abdominal x-ray showed a markedly distended stomach and a huge quantity of gas in right lower abdomen (). We performed continuous suction with a naso-gastric and rectal tube for decompression and observed the patient. The next day, abdominal distension progressively deteriorated and a follow-up laboratory examination revealed uncontrolled inflammation. A follow-up supine abdominal x-ray 24 hours after the decompression revealed fixed haustral gas formation despite decompression with gastric and rectal tube insertion (). Leukocytosis was improved, but C-reactive protein levels increased markedly to 34.11 mg/dL. An abdominal computed tomography scan without contrast enhancement scan showed dilated small bowel in the abdomen but no 'while' or 'bird peak' sign ().\nWe suspected intestinal obstruction as the primary cause of the symptoms and planned an explorative laparotomy. During the laparotomy, ischemic changes were seen throughout the cecum and part of the ascending colon. The cecum, which was not fixed at the retro-peritoneum, was dilated and twisted around the terminal ileum () and an approximately 20 cm length segmentectomy of the proximal ascending colon was completed with preserved ileocecal valve.\nThe patient was admitted to the intensive care unit and hospital ward for >2 months due to post-operative ileus. Approximately 3 months after the laparotomy, previous ileus signs had nearly disappeared and the patient's symptoms were much improved.