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A cohort of five cases with asymmetric conjoined twining and literature review

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Abstract

Purpose

Asymmetric conjoined twining (ACT) is a form of conjoined twining which is a rare malformation of monochorionic monoamniotic twin pregnancy. Most publications were single case reports. We reported a cohort of five cases with ACT from a single tertiary medical center and reviewed the case reports of ACT over the last decade to enrich the clinical research of this disease and summarized the clinical features of the disease.

Methods

We reviewed five cases of ACT admitted in Tianjin Children's Hospital from 17 March, 2008, through 7 March 2017. The cohort was analysed from general information, imaging manifestations, separation surgery, histopathological findings, outcome and follow-up. We searched the English literatures on case reports of ACT over the past decade from the PubMed database and presented details about the clinical characteristics, treatment, and prognosis of all cases.

Results

There were four males and one female in our cohort. Among the five cases, two parasites were located in epigastrium, two in rachis, and one in retroperitoneum (fetus in fetu, FIF). All of the parasites were separated successfully by operation in five cases and were confirmed to be ACT by histopathology reports. Four patients made an uneventful recovery except for one case of wound infection. All of them were doing well in follow-up. In the literature review, we found 41 cases of exoparasitic heteropagus twining (EHT) and 63 cases of FIF.

Conclusions

ACT is very rare and usually diagnosed by prenatal ultrasonography (US). Computed tomography (CT) and magnetic resonance imaging (MRI) examinations are essential imaging examinations before separation surgery to delineate the anatomical relationship between the autosite and the parasite. In general, the separation surgery of ACT is less complicated and the prognosis is better compared with the symmetric conjoined twining (SCT).

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Acknowledgements

Not applicable.

Funding

This work was supported by the National Natural Science Foundation of China (grant number 81771589) and the Program of Tianjin Science and Technology Plan (grant number 18ZXDBSY00170).

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Author notes

  1. Xiufang Zhi, Bo Hu and Xuwen Zhao contributed equally to this work.

Authors and Affiliations

  1. Graduate College of Tianjin Medical University, Tianjin, 300070, China

    Xiufang Zhi, Chunyu Gu & Linjie Pu

  2. Tianjin Children’s Hospital (Children’s Hospital of Tianjin University), Tianjin, 300134, China

    Xiufang Zhi, Chunyu Gu, Linjie Pu, Yulian Fang & Chunquan Cai

  3. Department of Neonatal Surgery, Tianjin Children’s Hospital, Tianjin, 300134, China

    Bo Hu & Xuwen Zhao

  4. Department of Radiology, Tianjin Children’s Hospital, Tianjin, 300134, China

    Jing Chen

  5. Tianjin Pediatric Research Institute, Tianjin, 300134, China

    Yulian Fang & Chunquan Cai

  6. Tianjin Key Laboratory of Birth Defects for Prevention and Treatment, Tianjin, 300134, China

    Yulian Fang & Chunquan Cai

  7. Department of Neurosurgery, Tianjin Children’s Hospital, No. 238 Longyan Road, Beichen District, Tianjin, 300134, China

    Chunquan Cai

Authors
  1. Xiufang Zhi
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  2. Bo Hu
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  8. Chunquan Cai
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Contributions

All authors contributed to the study conception and design. BH, XZ were responsible to design and conduct the study. Material preparation, data collection and analysis were performed by CG, LP, JC and YF. The first draft of the manuscript was written by XZ. CC was in charge of critical revisions and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

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Correspondence to Chunquan Cai.

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Zhi, X., Hu, B., Zhao, X. et al. A cohort of five cases with asymmetric conjoined twining and literature review. Pediatr Surg Int 38, 169–181 (2022). https://doi.org/10.1007/s00383-021-05006-w

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