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Hirschsprung's Disease-Related Giant Sigmoid Volvulus Complicated by Refractory Hypertension in an Elderly Man

Challenging differential diagnosis, Diagnostic / therapeutic accidents, Management of emergency care, Unexpected drug reaction , Rare disease, Educational Purpose (only if useful for a systematic review or synthesis)

Shaohan Wu, Xiaofang Sun, Yawei Yu, Yiyu Shen

(Department of General Surgery, The Second Affiliated Hospital of Jiaxing University, Jiaxing, Zhejiang, China (mainland))

Am J Case Rep 2018; 19:467-471

DOI: 10.12659/AJCR.908389

Published: 2018-04-20


BACKGROUND: Sigmoid volvulus (SV) is a life-threatening condition occasionally seen in adults. Adult Hirschsprung’s disease (HD)-related SV is rarely complicated by difficult-to-control hypertension. In this report we present the case of an elderly man with a rare constellation of HD, SV, and refractory hypertension.
CASE REPORT: An 82-year-old man had long-term constipation, moderate abdominal pain, and progressive abdominal distension. A CT scan revealed the typical “coffee bean sign”. Blood pressure was abnormal high. Subsequently, the patient's condition deteriorated. Therefore, he underwent a Hartmann's procedure. A giant and redundant sigmoid colon (length more than 60 cm, maximal diameter about 15 cm) was demonstrated to be the cause of SV during the process of surgery. Moreover, abdominal compartment syndrome caused by SV resulted in his high and refractory blood pressure (BP). Postoperative pathological results revealed HD in his sigmoid colon.
CONCLUSIONS: SV is rarely combined with conditions like refractory hypertension or HD among the elderly. Clinical features of SV typically present with long-term constipation, severe abdominal pain, and progressive abdominal distension. The “coffee bean sign” could be observed in imaging examinations. It is important to note that the management of SV is to relieve the obstruction and prevent recurrence, no matter which therapy is used in elderly patients with Hirschsprung's disease.

Keywords: Antihypertensive Agents, Hirschsprung Disease, Sigmoid Diseases



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