Bilateral ureteral reimplantation in a patient with an intraperitoneal ectopic bipenis:A case report

lNTRODUCTlON

Diphallia(penile duplication)is an extremely rare congenital urogenital anomaly,with an incidence of approximately one in 5-6 million births[1,2]Since Wecker reported the first case in 1609,reports on more than 100 cases have been published[3].Most of the available reports are about penile duplication in the perineum,which is visible to the naked eye.This is the first report of a patient with intraperitoneal ectopic bipenis to our knowledge,Mainly based on the literature review on PubMed database.

CASE PRESENTATlON

Chief complaints

A man in his 40s was admitted to the hospital with the chief complaint of hydronephrosis in both kidneys for 3 d.

History of present illness

Without significant lumbar or abdominal pain or bladder irritation.

Soon he agreed to “dangle,” sit on the side of the bed to build up sitting tolerance6, energy and balance. He agreed to “work” with physical therapy if I d return “to talk.”

History of past illness

He had previously undergone ureteral stent placement on the right side due to bilateral hydronephrosis in January 2020;however,ureteroscopic placement on the left side failed.Left ureterotomy and ureteral stent implantation were performed accordingly.The bilateral ureteral stents were removed 3 mo postoperatively.Other past history were all negative.

Personal and family history

The patient had no relevant personal and family history.

Physical examination

All authors declare that they have no potential conflicts of interest.

Laboratory examinations

Biochemical blood tests revealed no apparent abnormalities.

Imaging examinations

The patient was discharged with no postoperative events.Follow-up was performed for 2 mo,with no reported adverse events.The patient was advised to visit regularly,especially within 1-2 years after surgery.

FlNAL DlAGNOSlS

Postoperative pathology suggested that the urethra and urethral and penile corpus cavernosum tissues were visible in the specimen sent for examination,which was consistent with an ectopic penile morphology(Figure 3).

TREATMENT

The Goose Girl is finally gaining some autonomy. She is able to cast a simple spell, using her own magic, to save her hair from Curdken s attentions. This spell also brings her to the attention of the old king and helps him to recognize that she must be more than she appears. She is gaining some maturity through her adversity.

OUTCOME AND FOLLOW-UP

A previous ureteroscopy for bilateral hydronephrosis revealed bilateral ureteral malformation,derangement,and displacement of the seminal vesicle glands to the left side.Urological plain and enhanced computed tomography suggested bilateral hydronephrosis,ureteral dilatation,and ureteral confluence on the left side of the bladder,a malformation opening on the left side of the bladder wall(Figure 1),and an intrapelvic soft tissue shadow on the left side of the bladder,was considered a germline malformation,namely a bipenis(hidden penis in the abdominal cavity)(Figure 2).

DlSCUSSlON

Penile duplication is an extremely rare congenital anomaly that varies in presentation and location from case to case[1,4].Existing reports in the literature refer mainly to external penile duplication,whereas intra-abdominal ectopic penis has not been reported to date.

In general,ultrasonography helps to confirm the diagnosis by detecting the presence or absence of the corpus cavernosum or spongiosum and its number.It can also detect other associated abnormalities.However,this case was unique because the ectopic penis was located in the abdominal cavity.Furthermore,inexperienced sonographers may not always be able to identify this malformation.Magnetic resonance imaging(MRI)is a valuable method to accurately diagnose diphallia and associated malformations because T2-W images have the appropriate contrast resolution[1,10,11].One limitation of the study is that no further MRI was performed in this case to confirm the diagnosis.

There are many clinical classifications for these conditions,and some authors[2]classify them as follows:(1)True diphallia,(A)complete or(B)partial;(2)Bifid phallus:(A)partial(a)bifid glans(b)bifid shaft,or(B)complete.In 2017,Jesus[7]proposed a new simplified classification based on the clinical and surgical implications of each type:(1)True penile duplication(each duplicate penis has two corpora and one spongiosum);(2)Hemiphalluses(each penis has corpora and a hemiglans);(3)Pseudoduplication(normal penis with accessory penis-like tissue);and(4)Partial duplication(duplication involving only the distal penis).Our case had true penile duplication.True duplication is associated with other congenital defects[4,8,9].Our case also involved anomalies,such as bilateral ureteral opening malformations,bilateral testicular ectopia,and bilateral epididymal fusion.

The etiology of diphallia remains unclear;however,there are many possible embryological explanations.Duplication of the penis is caused by a lack of fusion of the paired mesodermal anlagen of the genital tubercle by the 15week of gestation[5].Many genetic alterations have been implicated in the development of supernumerary penises,some of which include genes that encode androgen receptors and are linked to the development of male external genitalia[3,4].Diphalia is not considered familial or hereditary[6].

Our case was first identified by radiologists,and the possibility of an intra-abdominal ectopic penis was considered based on plain and enhanced computed tomography of the urinary tract,which was dependent upon the radiologists' experience in reading the films and provided important information for the physicians.

In general,the associated malformations should be treated first[12].In the case of actual penile duplication,partial duplication,or pseudo-duplication,most surgeons choose to resect the hypoplastic duplicate penis,glans,or accessory penile-like tissues to maintain the main urethra[13].In this case,the ectopic penis was located in the abdominal cavity and crossed the left ureter anteriorly,which caused bilateral ureteral dilatation and hydronephrosis.Bilateral ureteral reimplantation is required to further protect renal function,and dissociation of the ectopic penis is also compulsory.

Their house caught fire and was speedily burnt to the ground, with all the splendid furniture, the books, pictures, gold, silver, and precious goods it contained; and this was only the beginning of their troubles

As the patient was a middle-aged male,he had no reproductive needs for the time being,but the status of the patient's sexual activity still requires further attention after surgery.

CONCLUSlON

To date,intra-abdominal ectopic bipenis has not been reported in humans.Penile duplication has a unique presentation in each patient,in which the position of the penis can be either ectopic or orthotopic.Depending on the corporal development and anatomy of the urethra,excision or reconstruction of the duplicate penis is required.

FOOTNOTES

The dwarfs said, If you will take care of our house, cook, make the beds, wash, sew, and knit, and if you will keep everything neat and clean, you can stay with us and you shall want for nothing

The patient provided informed consent for the publication of this case.

Physical examination showed normal external penile development,bilateral testes located on the left side of the scrotum,and a fused epididymis.The left-sided scrotal swelling that transilluminated upon application of light suggested a hydrocele.

Li YY and Zhu J designed the research study;Jia YT and Zhu J performed the research;Jia YT,Shi BL,and Zhang J Searched relevant literature and review;Jia YT and Zhu J sort out the information and wrote the manuscript;all authors have read and approve the final manuscript.

The authors have read the CARE Checklist(2016),and the manuscript was prepared and revised according to the CARE Checklist(2016).

These youngsters, dressed in sneakers so ragged6 that shoestrings7 seemed to be the only thing holding them together, presented a sharp contrast to our boys in their spiffy blue and gold uniforms and sparkling new wrestling shoes.

This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers.It is distributed in accordance with the Creative Commons Attribution NonCommercial(CC BYNC 4.0)license,which permits others to distribute,remix,adapt,build upon this work non-commercially,and license their derivative works on different terms,provided the original work is properly cited and the use is noncommercial.See: https://creativecommons.org/Licenses/by-nc/4.0/

Intraoperative exploration around the bladder revealed 8.0 cm × 2.0 cm columnar solid tissue outside the left posterior wall of the bladder,with tough texture,reaching down to the anterior rectal wall and extending upward to the pelvic wall(Supplementary Figure 1).No seminal vesicle was seen,and the lumen of the left ureter was dilated,with a diameter of about 1.5 cm,passing behind the columnar tissue.The right ureter was carried out in the cysto-rectal space,with a lumen diameter of approximately 1.0 cm;the pelvic columnar neoplasm was considered a repeat ectopic penis.Due to serious adhesions,partial excision of the pelvic mass was performed,with a length of approximately 2.0 cm excised from the middle section,the smooth muscle tissue visible inside,and a tubular channel visible in the middle.The 7-0 silk sutures were wrapped and sutured at the stump,whereas the right and left ureters were loosened.Next,the ureters were cut short at the lowest point and reimplanted on the left and right anterior walls of the bladder,respectively,while bilateral ureteral stents were left in place.

China

19.Goose: A goose is usually associated with women and the household (Biedermann 156). A foolish person is sometimes called a goose, or a silly goose (Evans 476). Dummling could be considered a silly goose .Jack Zipes considers the goose a phallic symbol associated with magic powers (The Great Fairy Tale 678) when used in humorous stories.The goose can also be associated with perfection and the Holy Ghost (Barley 265). Return to place in story.

Ya-Tao Jia 0000-0003-1280-0366;Bao-Lei Shi 0000-0003-1280-0258;Jie Zhang 0000-0003-1280-5768;Ying-Yi Li 0000-0003-1280-0237;Jiang Zhu 0000-0002-9236-9811.

Then he asked if she thought she could be happy in his palace; and Beauty answered that everything was so beautiful that she would be very hard to please if she could not be happy

Liu JH

A

Quick, away with him, and off with his head! So the false slave was put to death, that none might follow in his footsteps, and the wedding feast was held, and the hearts of all rejoiced that the true bridegroom had come at last

Liu JH