1130-0108/2016/108/11/742-746
REVISTA ESPAÑOLA DE ENFERMEDADES DIGESTIVAS
© Copyright 2016. SEPD y © ARÁN EDICIONES, S.L.
REV ESP ENFERM DIG
2016, Vol. 108, N.º 11, pp. 742-746
CASE REPORTS
ABSTRACT
Hirschsprung’s disease is characterized by absence of ganglion
cells in submucosal and myenteric plexus of distal bowel. Most cases
become manifest during the neonatal period, but in rare instances,
this disease is initially diagnosed in adult age. It usually presents as
severe constipation with colonic dilatation proximal to the agangli-
onic segment. The treatment is surgical, removing the aganglionic
segment and restoring continuity of digestive tract. The disease
rarely presents as an acute intestinal obstruction. We report a case
not previously diagnosed, which presented as a massive colonic
dilatation with a maximum diameter of 44 cm, with imminent risk
of drilling that forced to perform an emergency surgery. We include
a review of existing literature.
Key words: Hirschsprung disease. Megacolon. Intestinal obstruc-
tion.
INTRODUCTION
Hirschsprung’s disease (HD) is a malformation of the
large intestine characterized by the absence of ganglion
cells in submucosal and myenteric plexus, which pro-
duces a functional obstruction and dilatation proximal
to the affected segment (1). This congenital disease is
usually detected in neonatal period. It is rarely diag-
nosed in adult age. Approximately 300 cases have been
reported in literature. These patients tend to have milder
forms of the disease, in which hypertrophy of innervated
proximal colon compensates the functional obstruction
by lack of innervation of the distal area (2). We report a
case of a 54-year-old male patient that was admitted in
the Emergency Department because of a massive mega-
colon that required a total colectomy with ileum-rectal
anastomosis.
CASE REPORT
A 54-year-old male was admitted in the Emergency
Department by generalized abdominal pain accompanied
by anorexia of several days duration. Previous history of
meningioma operated in 1998, intestinal resection in child-
hood (unknown etiology), chronic constipation, smoker
of 20 cigarettes per day and moderate drinker. The patient
reported difficulty for expelling gases, although he expelled
stools without pathological products. No nausea or vomit-
ing. No fever. Exploration showed a hemodynamically sta-
ble patient, eupneic with blood pressure 138/97 and heart
rate 100 beats per minute. Abdomen was very distended and
tympanic, with previous laparotomy scar, painful on palpa-
tion diffusely. Peritoneal irritation was not found. Peristal-
sis was present. Abdominal X-rays showed a dilated colon
occupying abdominal cavity. Laboratory studies revealed
leukocytosis with left deviation and elevated C-reactive pro-
tein. A CT scan was performed, showing a massive dilatation
of sigmoid colon, which compresses liver to the right upper
quadrant, stomach into the left upper quadrant and bowel
loops into the right flank (Figs. 1 and 2). A transition zone
was appreciated in distal sigmoid colon. Rectum had a nor-
mal size. Peripheral aerial imagery was found in the dilated
segment, gas-like wall, which is thickened in this segment.
Given these findings, emergency surgery was decided. A
midline laparotomy was performed, finding a massive dila-
tation of sigmoid colon, attaining a diameter of 44 cm at its
more dilated portion. The rest of the colon was also dilat-
ed. Rectum had normal caliber. It was decided to perform
a total colectomy with ileum-rectal anastomosis. After 24
hours, the patient presented hemodynamic instability, with
severe abdominal pain. CT scan was performed, showing
Received: 18-05-2015
Accepted: 04-09-2015
Correspondence: José Antonio López-Ruiz. Emergency Surgery Unit.
Department of General and Gastrointestinal Surgery. Complejo Hospitalario
Virgen Macarena-Virgen del Rocío. Av. Doctor Fedriani, 3. 41007 Sevilla,
Spain
e-mail: wurdalach@gmail.com
López-Ruiz JA, Tallón-Aguilar L, Sánchez-Moreno L, López-Pérez J, Pareja-
Ciuró F, Oliva-Mompeán F, Padillo-Ruiz J. Hirschsprung disease with debut
in adult age as acute intestinal obstruction: case report. Rev Esp Enferm Dig
2016;108(11):742-746.
DOI: 10.17235/reed.2016.3841/2015
Hirschsprung disease with debut in adult age as acute intestinal obstruction:
case report
José Antonio López-Ruiz, Luis Tallón-Aguilar, Laura Sánchez-Moreno, José López-Pérez, Felipe Pareja-Ciuró, Fernando Oliva-Mompeán
and Javier Padillo-Ruiz
Emergency Surgery Unit. Department of General and Gastrointestinal Surgery. Hospital Universitario Virgen Macarena. Seville, Spain