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Epigastric heteropagus conjoined twins: two case studies and associated DNA analysis.

Xie JT, Zhou L, Yang ZL, Sun HY - Clinics (Sao Paulo) (2012)

View Article: PubMed Central - PubMed

Affiliation: Sun Yat-sen University, The First Affiliated Hospital of Guangzhou, Department of Pediatric Surgery, People's Republic of China.

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Heteropagus conjoined twins are asymmetrically conjoined twins, in which one twin is nearly anatomically normal (autosite), and the other twin remains anatomically incomplete (parasite) but physically attached to the autosite... Epigastric heteropagus conjoined twins (EHTs), referring to a parasitic twin who is attached to the epigastrium of the autosite, are extremely rare... The parasite possessed two upper limbs, two lower limbs, a pelvis, and a well-developed scrotum and penis, which produced urine discharge (Figure 1-A)... However, the limbs of the parasite had neither active movement nor response to tactile stimulus... The parasitic twin was attached to the epigastrium of the autosite, in whom an infected omphalocele and a large ventral hernia were found (Figure 2-A)... Omphaloceles were present below the cartilage by which the twins were attached in both of our cases... Many studies have considered omphaloceles to be commonly associated anomalies in EHTs... Tongsin et al. reported four cases of EHTs, three of which were associated with omphaloceles... The large hernia rendered the repair of the abdominal wall particularly difficult because the simultaneous repair of the hernia and removal of the parasitic twin would have significantly increased the intra-abdominal pressure and could have caused ICS... Therefore, we delayed the hernia repair until the twentieth month after birth... VYPRO II mesh (Ethicon) was used to successfully repair the hernia... Despite the unusual clinical appearance and pathogenesis associated with EHTs, EHT patients usually have a good prognosis following a successful separation surgery... However, early diagnosis, diligent prenatal management, and selection of the proper route of delivery are critical to the success in treating EHT patients.

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(A) Large hernia of the abdominal wall (H), omphalocele (O) and limbs (L) of the parasitic twin in case 2. (B) View of the hernia in case 2. The parasitic twin was excised, and the omphalocele was repaired. (C) The pelvis of the parasitic twin in case 2 contained one small kidney and a cyst. (D) The hernia in case 2 was repaired using VYPRO II mesh. (E) Condition of the repaired ventral hernia in case 2 at 14 days after surgery. (F) The autosite in case 2 during the twenty-third month after the operation to remove the parasitic twin.
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f2-cln_67p527: (A) Large hernia of the abdominal wall (H), omphalocele (O) and limbs (L) of the parasitic twin in case 2. (B) View of the hernia in case 2. The parasitic twin was excised, and the omphalocele was repaired. (C) The pelvis of the parasitic twin in case 2 contained one small kidney and a cyst. (D) The hernia in case 2 was repaired using VYPRO II mesh. (E) Condition of the repaired ventral hernia in case 2 at 14 days after surgery. (F) The autosite in case 2 during the twenty-third month after the operation to remove the parasitic twin.

Mentions: Case 2: The second patient was also a 1-day-old FTND male. The total weight of the autosite and parasite was 3.4 kg. The parasitic twin consisted of 2 immobile lower limbs, buttocks, perineum, and masculine genitalia. The parasitic twin was attached to the epigastrium of the autosite, in whom an infected omphalocele and a large ventral hernia were found (Figure 2-A). The autosite was generally stable, and no respiratory distress was observed. The omphalocele was repaired on the eighth day after birth, and the parasitic twin was successfully removed on the twentieth day after birth (Figure 2-B). Similar to case 1, the parasite was attached to the sternum of the autosite through a tract of cartilage. The pelvis of the parasite contained one small kidney and a cyst (Figure 2-C). Skeletal muscle in the limbs of the parasitic twin was absent. The large hernia was not repaired during the separation of the twins to avoid “intra-abdominal compartment syndrome” (ICS) (2). The hernia was eventually repaired using Ethicon VYPRO II mesh (Ethicon, Somerville, N.J., USA) (Figure 2-D) when the autosite reached 20 months of age. The patient recovered from the hernia surgery without complications (Figure 2-E). DNA analysis was performed on the patient in case 2. DNA samples from the skin and hair of the autosite and from the skin and kidney of the parasitic twin were extracted using the rapid Chelex-100 method. A sex locus (amelogenin gene) and 15 STR loci were amplified using the Powerplex™ 16 system (Promega). PCR products were separated using an ABI3100 genetic analyzer (Applied Biosystems Inc.) and were genotyped using GeneMapper® 3.7 (Applied Biosystems Inc.). DNA analysis revealed that the parasitic twin in the second case had an identical genotype to the autosite, which indicated that they were of monozygotic origin (Figure 3). The autosite was followed up for 30 months, and Figure 2-F shows the patient in case 2 at 23 months after the surgical removal of the parasitic twin. The omphalocele and large hernia of the abdominal wall were closed completely in the patient in case 2.


Epigastric heteropagus conjoined twins: two case studies and associated DNA analysis.

Xie JT, Zhou L, Yang ZL, Sun HY - Clinics (Sao Paulo) (2012)

(A) Large hernia of the abdominal wall (H), omphalocele (O) and limbs (L) of the parasitic twin in case 2. (B) View of the hernia in case 2. The parasitic twin was excised, and the omphalocele was repaired. (C) The pelvis of the parasitic twin in case 2 contained one small kidney and a cyst. (D) The hernia in case 2 was repaired using VYPRO II mesh. (E) Condition of the repaired ventral hernia in case 2 at 14 days after surgery. (F) The autosite in case 2 during the twenty-third month after the operation to remove the parasitic twin.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3351264&req=5

f2-cln_67p527: (A) Large hernia of the abdominal wall (H), omphalocele (O) and limbs (L) of the parasitic twin in case 2. (B) View of the hernia in case 2. The parasitic twin was excised, and the omphalocele was repaired. (C) The pelvis of the parasitic twin in case 2 contained one small kidney and a cyst. (D) The hernia in case 2 was repaired using VYPRO II mesh. (E) Condition of the repaired ventral hernia in case 2 at 14 days after surgery. (F) The autosite in case 2 during the twenty-third month after the operation to remove the parasitic twin.
Mentions: Case 2: The second patient was also a 1-day-old FTND male. The total weight of the autosite and parasite was 3.4 kg. The parasitic twin consisted of 2 immobile lower limbs, buttocks, perineum, and masculine genitalia. The parasitic twin was attached to the epigastrium of the autosite, in whom an infected omphalocele and a large ventral hernia were found (Figure 2-A). The autosite was generally stable, and no respiratory distress was observed. The omphalocele was repaired on the eighth day after birth, and the parasitic twin was successfully removed on the twentieth day after birth (Figure 2-B). Similar to case 1, the parasite was attached to the sternum of the autosite through a tract of cartilage. The pelvis of the parasite contained one small kidney and a cyst (Figure 2-C). Skeletal muscle in the limbs of the parasitic twin was absent. The large hernia was not repaired during the separation of the twins to avoid “intra-abdominal compartment syndrome” (ICS) (2). The hernia was eventually repaired using Ethicon VYPRO II mesh (Ethicon, Somerville, N.J., USA) (Figure 2-D) when the autosite reached 20 months of age. The patient recovered from the hernia surgery without complications (Figure 2-E). DNA analysis was performed on the patient in case 2. DNA samples from the skin and hair of the autosite and from the skin and kidney of the parasitic twin were extracted using the rapid Chelex-100 method. A sex locus (amelogenin gene) and 15 STR loci were amplified using the Powerplex™ 16 system (Promega). PCR products were separated using an ABI3100 genetic analyzer (Applied Biosystems Inc.) and were genotyped using GeneMapper® 3.7 (Applied Biosystems Inc.). DNA analysis revealed that the parasitic twin in the second case had an identical genotype to the autosite, which indicated that they were of monozygotic origin (Figure 3). The autosite was followed up for 30 months, and Figure 2-F shows the patient in case 2 at 23 months after the surgical removal of the parasitic twin. The omphalocele and large hernia of the abdominal wall were closed completely in the patient in case 2.

View Article: PubMed Central - PubMed

Affiliation: Sun Yat-sen University, The First Affiliated Hospital of Guangzhou, Department of Pediatric Surgery, People's Republic of China.

AUTOMATICALLY GENERATED EXCERPT
Please rate it.

Heteropagus conjoined twins are asymmetrically conjoined twins, in which one twin is nearly anatomically normal (autosite), and the other twin remains anatomically incomplete (parasite) but physically attached to the autosite... Epigastric heteropagus conjoined twins (EHTs), referring to a parasitic twin who is attached to the epigastrium of the autosite, are extremely rare... The parasite possessed two upper limbs, two lower limbs, a pelvis, and a well-developed scrotum and penis, which produced urine discharge (Figure 1-A)... However, the limbs of the parasite had neither active movement nor response to tactile stimulus... The parasitic twin was attached to the epigastrium of the autosite, in whom an infected omphalocele and a large ventral hernia were found (Figure 2-A)... Omphaloceles were present below the cartilage by which the twins were attached in both of our cases... Many studies have considered omphaloceles to be commonly associated anomalies in EHTs... Tongsin et al. reported four cases of EHTs, three of which were associated with omphaloceles... The large hernia rendered the repair of the abdominal wall particularly difficult because the simultaneous repair of the hernia and removal of the parasitic twin would have significantly increased the intra-abdominal pressure and could have caused ICS... Therefore, we delayed the hernia repair until the twentieth month after birth... VYPRO II mesh (Ethicon) was used to successfully repair the hernia... Despite the unusual clinical appearance and pathogenesis associated with EHTs, EHT patients usually have a good prognosis following a successful separation surgery... However, early diagnosis, diligent prenatal management, and selection of the proper route of delivery are critical to the success in treating EHT patients.

Show MeSH
Related in: MedlinePlus