Abnormally Shaped Arteries in the Intestine of Children with Hirschsprungʼs Disease

Taguchi, Tomoaki; Suita, Sachiyo; Hirata, Yoshihiro; Hirose, Ryoji; Yamada, Takaharu; Toyohara, T.

doi:10.1097/00005176-199402000-00013

Cited by 19 publications

(13 citation statements)

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“…Spontaneous perforation of the small bowel has been described in children with this disease [32]. In this case it is highly unlikely that this underlying disorder contributed significantly to the bowel perforation.…”

Section: Commentmentioning

confidence: 76%

Small bowel perforation after thermoregulated radiofrequency endometrial ablation: a case report

Cosyns

Poots

Lind³

2006

Gynecol Surg

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During the past decades, numerous hysteroscopic ablation techniques have been developed for the treatment of menorrhagia, all conferring relatively comparable success rates and low complication incidences. We here report an unusual, adverse, post-operative, complication of the Vesta thermoregulated radiofrequency endometrial ablation system in a 34-year-old nulliparous woman with dysfunctional uterine bleeding. Fourteen days after the procedure she presented with acute abdominal pain. At laparotomy, a small bowel perforation was identified, and the entire uterus was found to be necrotic, necessitating a total hysterectomy. This is the first report of a severe complication of this endometrial ablation system in the absence of uterine perforation. We propose that minimal myometrial thickness should be taken into consideration to improve the safety of thermoregulated radiofrequency endometrial ablation.

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Section: Commentmentioning

confidence: 76%

Small bowel perforation after thermoregulated radiofrequency endometrial ablation: a case report

Cosyns

Poots

Lind³

2006

Gynecol Surg

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“…We wished to share this experience with an idea to enhance the awareness regarding possibility of existence of this extremely rare association of congenital anomalies. A possible explanation for the coexistence of these two conditions would be an ischemic insult occurring during embryogenesis and interfering both with the gut canalization and neural crest migration [6,7]. …”

Section: Discussionmentioning

confidence: 99%

Hirschsprung’s Disease with Bowel Duplication A Rarest of Rare Anomalous Association

Ah¹,

Ta²,

Fa³

et al. 2017

JPNC

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Submit Manuscript | http://medcraveonline.com infant developed progressive abdominal distention while under observation in the neonatal ICU. On examination, the infant looked generally well, maintaining on room air, vital signs were normal and no dismorphic features. Abdomen was distended all over, there was no visible peristalsis or visible bowel loops with normal overlying skin. It was soft and lax, with no palpable mass, but had exaggerated bowel sounds. Digital rectal examination revealed a normal anal orifice with normal muscle tone. Baby passed little sticky meconium after rectal stimulation, but no gush of gas or stool on withdrawal of finger. A preliminary clinical impression of meconium plug syndrome was made, and conservative treatment started including fleet enema, but unfortunately, the condition of the patient did not improve in spite of repeated enemas. Blood counts, Hemoglobin, serum chemistry, renal functions were within normal values. Liver function showed unconjugated hyperbilirubinemia, but other parameters were normal. Plain abdominal x ray revealed dilated bowel loops all over the abdomen, no air fluid levels, no free intraperitoneal gas, no intramural gas, and no evidence of any mass lesion. However it revealed diffuse gaseous distention of colon, more prominent on the left side. Gastrograffin contrast enema study was undertaken, for diagnostic as well as therapeutic purposes, due to suspicion of meconium plug syndrome. It revealed classic cone shaped narrowing in recto sigmoid area, with proximal dilatation of bowel and distal narrowing suggesting the presence of Hirschsprung's disease (Figure 1).The dye was retained in the bowel for more than 24 hours. Baby kept NPO and was put on TPN, frequent OGT aspiration. Baby was monitored continuously and prepared for surgery. Repeated x ray of the abdomen revealed an increase in bowel distention and appearance of air fluid levels. Exploratory laparotomy was performed on day six of life. Intraoperatively, we found dilated large colon, with transitional zone at rectosigmoid level, and distal collapsed bowel (Figure 2). Multiple full thickness biopsies were taken from the collapsed, dilated bowel loops as well as the transitional zone. The exploration of the rest of the bowel revealed the presence of a moderate sized mesenteric cyst in the mid ileum (Figure 3). The cyst was quite tense, thick walled, stretching the adjoining loop of ilium and causing partial intestinal obstruction. In view of these finding, a wedge resection of the cystic lesion along with the affected bowel loop was undertaken, and end to end anastomosis of the ileum was performed. A divided colostomy was performed just above the transition zone and the abdomen closed. AbstractHirschsprung's Disease is an uncommon cause of functional intestinal obstruction in neonates, especially females. Duplication of bowel is also a rare cause of mechanical obstruction in children at birth. An association of these two congenital anomalies is not reported in the available English language literat...

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“…[9] Taquchi et al in their study noted abnormally shaped arteries in the intestine of children with Hirschsprung's disease and based their aetiological consideration to ischaemic theory. [10] Each of the conditions is responsible for morbidity and mortality in paediatric surgical practice. The coexistence of the two conditions may aggravate or increase the morbidity and mortality in children.…”

Section: Discussionmentioning

confidence: 99%

Anorectal malformation coexisting with Hirschsprung′s disease: A report of two patients

Lukong

Mshelbwala

Anumah

et al. 2012

Afr J Paediatr Surg

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Anorectal Malformation (ARM) and Hirschsprung's Disease (HD) are common causes of congenital intestinal obstruction in children. Simultaneous occurrence of both conditions is rare. Few have been reported in Europe and Asia, but we have no knowledge so far of such report from Nigeria. We present two patients managed in our centre to highlight the challenges of management of this uncommon coexistence. The first patient was a 5-year-old girl who was referred to us with intestinal obstructive symptoms despite an apparently adequate sized ectopic anus. She had colostomy and rectal biopsy, which confirmed HD. She had corrective surgery performed through a posterior sagittal approach. She did well post operatively. The second patient was a 3-year-old girl who presented with features of intestinal obstruction, had laparatomy and was also referred to us. It was observed in the referral hospital during laparatomy, to have features of HD and rectal atresia intraoperatively. She had colostomy done and rectal biopsy performed at the same time, which confirmed the diagnosis of HD. She had simultaneous correction of both conditions through a posterior sagittal approach. She was in good condition at follow up. It was therefore recommended that a high index of suspicion of HD, should be entertained while managing patients with anorectal malformation.

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Abnormally Shaped Arteries in the Intestine of Children with Hirschsprungʼs Disease

Cited by 19 publications

References 0 publications

Small bowel perforation after thermoregulated radiofrequency endometrial ablation: a case report

Small bowel perforation after thermoregulated radiofrequency endometrial ablation: a case report

Hirschsprung’s Disease with Bowel Duplication A Rarest of Rare Anomalous Association

Anorectal malformation coexisting with Hirschsprung′s disease: A report of two patients

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