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一例极为罕见的双阴茎合并肛门直肠畸形病例。

A very rare case of diphallia with anorectal malformation.

作者信息

Habib Murad, Bajwa Hajra Fazeelat, Abbas Muhammad, Chaudhary Muhammad Amjad

机构信息

Department of Paediatric Surgery, The Children's Hospital, Pakistan Institute of Medical Sciences, Islamabad, Pakistan.

Akbar Niazi Teaching Hospital, Islamabad, Pakistan.

出版信息

Int J Surg Case Rep. 2023 Apr;105:107980. doi: 10.1016/j.ijscr.2023.107980. Epub 2023 Mar 16.

DOI:10.1016/j.ijscr.2023.107980
PMID:36933406
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10031018/
Abstract

INTRODUCTION AND IMPORTANCE

Diphallia is extremely rare urological anomaly with reported incidence of 1 in 5-6 million live births. It can present as complete or incomplete diphallia. In most of the cases it is associated with complex urological, gastrointestinal or anorectal malformations.

CASE PRESENTATION

We report here case of a newborn who was presented to us at 1st day of life with diphallia and anorectal malformation. He had true diphallia with two separate uretheral orifices. Both phalluses were uncircumcised, phallus 1 was 2.5 cm in length while phallus 2 was 1.5 cm. Both phalluses had normal shaped glans with uretheral opening located at the normal place He had a single scrotum with two midline raphe and well-formed rouge. He was passing urine from both orifices. His ultrasonography of urological system showed two ureters and a single hemi bladder. He was admitted and operated upon and a sigmoid divided colostomy was constructed. Per-operatively congenital pouch colon (type 4) was identified. His post-operative recovery was uneventful. The patient was discharged on second post-operative day and called for follow up.

CLINICAL DISCUSSION

Diphallia is a rare congenital anomaly, which means two structurally and anatomically separate phalluses. Complete Duplication in Diphallia presents the kind of Diphallia in which both the phalluses have two corpus cavernosum and only one corpus spongiosum. As diphallia presents with a spectrum of diseases; therefore, it requires a multidisciplinary approach. A case of Diphallia may well present with complex urogenital, gastrointestinal or anorectal malformations. As in our case the patient had Diphallia with anorectal malformation. Hence he was operated upon and a sigmoid colostomy was constructed.

CONCLUSION

Diphallia is a very rare congenital anomaly which can occur in association with anorectal malformations. Management of such cases should be individualized depending upon the spectrum of disease.

摘要

引言与重要性

双阴茎是一种极为罕见的泌尿系统异常,据报道其在活产婴儿中的发病率为1/500万至600万。它可表现为完全性或不完全性双阴茎。在大多数情况下,它与复杂的泌尿系统、胃肠道或肛门直肠畸形相关。

病例报告

我们在此报告一例新生儿,其在出生第一天因双阴茎和肛门直肠畸形前来就诊。他患有真性双阴茎,有两个独立的尿道口。两个阴茎均未行包皮环切术,阴茎1长2.5厘米,而阴茎2长1.5厘米。两个阴茎的龟头形状正常,尿道口位于正常位置。他有一个单一的阴囊,有两条中线嵴和发育良好的肉阜。他从两个尿道口排尿。其泌尿系统超声检查显示有两条输尿管和一个半膀胱。他入院后接受了手术,构建了乙状结肠造口术。术中发现先天性袋状结肠(4型)。他术后恢复顺利。患者在术后第二天出院,并安排了随访。

临床讨论

双阴茎是一种罕见的先天性异常,即两个在结构和解剖上相互独立的阴茎。双阴茎中的完全重复表现为两个阴茎均有两个海绵体和仅一个尿道海绵体的那种双阴茎。由于双阴茎伴有一系列疾病;因此,它需要多学科方法。一例双阴茎病例很可能伴有复杂的泌尿生殖系统、胃肠道或肛门直肠畸形。如我们的病例中,患者患有双阴茎合并肛门直肠畸形。因此,他接受了手术并构建了乙状结肠造口术。

结论

双阴茎是一种非常罕见的先天性异常,可与肛门直肠畸形相关。此类病例的管理应根据疾病谱进行个体化。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6860/10031018/e80cddd8094e/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6860/10031018/0f958d35582e/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6860/10031018/178129f6563c/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6860/10031018/e80cddd8094e/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6860/10031018/0f958d35582e/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6860/10031018/178129f6563c/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6860/10031018/e80cddd8094e/gr3.jpg

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