Caudal duplication syndrome: imaging evaluation of a rare entity in an adult patient

Abstract

Several theories have been put forth to explain the complex yet symmetrical malformations and the myriad of clinical presentations of caudal duplication syndrome. Hereby, reported case is a 28-year-old female, gravida 2 para 2, with congenital caudal malformation who has undergone partial reconstructive surgeries in infancy to connect her 2 colons. She presented with recurrent left lower abdominal pain associated with nausea, vomiting, and subsequent feculent anal discharge. Imaging reveals duplication of the urinary bladder, urethra, and colon with with cloacal malformations and fistulae from the left-sided cloaca, uterus didelphys with separate cervices and vaginal canals, right-sided aortic arch and descending thoracic aorta, and dysraphic midline sacrococcygeal defect. Hydronephrosis of the left kidney with left hydroureter and inflammation of one of the colons were suspected to be the cause of the patient’s acute complaints. She improved symptomatically over the course of her hospitalization stay with conservative treatments. The management for this syndrome is individualized and may include surgical intervention to fuse or excise the duplicated organs.

Polymelia and duplication of the descending colon in a Poodle dog - a case report

“…One of the most common and accepted is the incomplete separation of mono-ovular twins or conjoined twinning as a result of the misexpression of one or more of the distal hOX genes, potentially hOX10 and hOX11 (Shah and JOShI, 2006;hU et al, 2016). In this case report, since the animal presented with a duplication of the descending colon and one anal orifice, it was presumed that the animal could adapt to its morphological condition and live a healthy life.…”

“…In humans, intestinal duplications are very rare (kUMe et al, 2007;LIaQat et al, 2014;hU et al, 2016); even though it may occur from the oral cavity to the anus (heISS, 1996;LIaQat et al, 2014), descending colon duplication is e. r. Daneze and F. B. J. Brasil: Polymelia and duplication of the descending colon in a Poodle dog extremely rare (SaLVaDOr et al, 1996). the occurrence of enteric duplication varies greatly and is usually mistaken for other gastrointestinal tract pathologies.…”

“…Sometimes its presentation is confusing because patients may present with palpable abdominal or inguinal pain and/or an abdominal mass (LIaQat et al, 2014). Colon duplication may be discovered accidentally while performing a colostomy (LIaQat et al, 2014;hU et al, 2016) or contrast radiography (kUMe et al, 2007).…”

“…the embryological explanation of the duplication of the colon is controversial and many theories have been proposed (SterN and WarNer, 2000;hU et al, 2016). One of the most common and accepted is the incomplete separation of mono-ovular twins or conjoined twinning as a result of the misexpression of one or more of the distal hOX genes, potentially hOX10 and hOX11 (Shah and JOShI, 2006;hU et al, 2016).…”

“…Intestinal duplication is rare in humans and animals, and is often associated with other major abnormalities of the spine, bladder and genitalia (LONGhOFer et al, 1991;arthUr et al, 2003;kUMe et al, 2007;LIaQat et al, 2014;hU et al, 2016). In animals, there have been three case reports in dogs (JOhNStON et al, 1989;LONGhOFer et al, 1991;arthUr et al, 2003) and one in cats (kraMer et al, 2007).…”