Caudal Duplication Syndrome—Report of a Case and Review of Literature

Swaika, Sweta; Basu, Sujit; Bhadra, Ram Chandra; Sarkar, Ruchirendu; Maitra, Somnath

doi:10.1007/s12262-013-0838-z

Cited by 10 publications

(8 citation statements)

References 5 publications

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“…Duplication of the external genitalia 1,2,4,9–14 and bladder 1–4,9–13,15,16 have been reported within several case studies and appear to be common presentations of the syndrome. Additional reported anomalies include duplication of the anus, 1–4,9,12 colon, 1,2,4,9–14 urethra, 2,4,7,10,11,16 and uterus. 3,4,10,17 Associated anomalies such as imperforate anus, 10,14,17 omphalocele, 2,10 lipomeningocele, 12 myelomeningocele, 2 and lipomyomeningocele 18 have also been documented.…”

Section: Discussionmentioning

confidence: 95%

A Rare Sonographic Case of Caudal Duplication Versus Polymelia

Scholl

Thacker

2020

Journal of Diagnostic Medical Sonography

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Caudal duplication is a rare condition that results in varying degrees of duplication of organs within the gastrointestinal, genitourinary, reproductive, spinal, and vertebral systems. Polymelia is a rare congenital defect that presents as supernumerary limbs. This case report describes a fetus with characteristics of both conditions but does not present with the classic scenario of either condition.

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Section: Discussionmentioning

confidence: 95%

A Rare Sonographic Case of Caudal Duplication Versus Polymelia

Scholl

Thacker

2020

Journal of Diagnostic Medical Sonography

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“…Bremer has postulated that under normal circumstances, portions of the intestinal tract become obliterated at 6th-7th week when vacuoles appear in these cell masses and subsequently coalesce to reconstitute a single lumen. However, if one or more of the vesicle becomes pinched off, a second lumen is created [7] , [15] .…”

Section: Discussionmentioning

confidence: 99%

“…The urinary bladder septum is removed to create a single chamber. The duplicated colons are usually not fused and have separate blood supply, and either stripping of mucosa or resection of duplicated colon and rectum can be undertaken [15] . If left untreated, some common complications associated with duplicated hindgut include constipation, obstruction, volvulus, and neoplastic changes [10] .…”

Section: Discussionmentioning

confidence: 99%

“… [13] demonstrated that patients are capable of successful vaginal delivery after surgical repair of cloacal malformation. Similar surgery can be performed in male patients to remove one of the hemiphalluses for cosmetic reasons [7] , [15] . Although an overall good surgical outcome is reported, the controversy in treatment is mainly centered around whether the risks and burdens of potential complications of surgery outweigh the psychological impact on the patient’s self-esteem and quality of life [6] .…”

Section: Discussionmentioning

confidence: 99%

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

Browning

Bishop

2016

Radiology Case Reports

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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.

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“…The importance of active involvement of family members and the patient itself is well recognized, but not always available. Parents must cope with the difficulty of accepting and adjusting to their child's condition, coordinate appointments with different healthcare providers, find information about their child's malformations, manage the financial demands of long-term medical care, and locate appropriate care centers [21,22]. Factors such as low income, socially disadvantaged groups, and inaccessible educational materials due to rarity of the malformations, limited access to information or lack of support groups can negatively influence adherence to the treatment plan.…”

Section: Support Groups and Quality Of Lifementioning

confidence: 99%

Caudal Duplication Syndrome Systematic Review—A Need for Better Multidisciplinary Surgical Approach and Follow-Up

et al. 2020

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Background and Objectives: Caudal duplication syndrome is a rare association of anatomical anomalies describing duplication of the hindgut, spine, and uro-genital structures, leading to varied clinical presentations. The current literature focuses on case reports which describe the embryological etiology and anatomical spectrum of the condition giving little attention to the surgical preparation, the need for a well-structured follow-up program, or the transition into adult healthcare of these complex patients. No reviews have been published regarding this complex pathology. Materials and Methods: A review of caudal duplication syndrome cases was done to assess the range of the clinical malformations, timing, and types of surgical interventions. Inconsistencies in multidisciplinary care, follow-up, and risk events were described. Results: Hindgut duplication always involved the anorectal region. Anorectal malformations were evenly distributed as unilateral and bilateral. Colon duplication extended from the anal region to the transverse colon or ascending colon in most of the cases and less to terminal. In females, genital duplication was present in all cases. The follow-up period varied between 3 months and 12 years. In all adult females, the motive of presentation was related to pregnancy (complications after successful delivery, fertility evaluation) or late complications (fecalith obstruction of the end-to-side colon anastomosis, repeated UTIs with renal scarring). Conclusions: Complex malformations affecting multiple caudal organs may have a strong impact in many aspects of the long-term quality of life; therefore, patients with caudal duplication syndrome need increased awareness and joined multidisciplinary treatment.

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Caudal Duplication Syndrome—Report of a Case and Review of Literature

Cited by 10 publications

References 5 publications

A Rare Sonographic Case of Caudal Duplication Versus Polymelia

A Rare Sonographic Case of Caudal Duplication Versus Polymelia

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

Caudal Duplication Syndrome Systematic Review—A Need for Better Multidisciplinary Surgical Approach and Follow-Up

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