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. 2022 Jun 9;92(9):2053–2065. doi: 10.1111/ans.17846

Diphallia: literature review and proposed surgical classification system

Dylan John Kendrick 1,2,✉, Roy Mark Kimble 1,2,3
PMCID: PMC9539879  PMID: 35678221

Abstract

Background

Diphallia occurs once in 5–6 million births, with no two patients presenting with the same anatomical variation. Here we discuss a review of diphallia case reports, as well as present a new surgical classification system based on the soft tissue composition of the two phalluses, the anatomy of the urethra present within the most normal phallus and the bladder configuration.

Methods

Eighty‐seven diphallia case reports were collected and analysed, excluding those presented in animals and articles that were non‐English, with the results compiled to provide an in‐depth reference of the specific anatomy found in diphallia patients and the associated abnormalities.

Results

Our proposed classification system was then applied to each patient and the most common configuration base on our classification system presented, along with commonly seen associated anomalies.

Conclusion

The reviewed cases represent a subset of the most unique diphallia patients; thus, several cases may be left unreported. Future reports can then be categorized, aiding as a reference, and potentially building on the classification, should the patient not fit into a specific group, leading to an expansion of the classification system.

Keywords: classification, congenital anomaly, diphallia, duplicate penis, urethral duplication

Proposed surgical classification system to provide a detailed and specific category for diphallia based on the soft tissue composition of the phallus, the urethra present in the most normal phallus and the bladder configuration.

graphic file with name ANS-92-2053-g005.jpg

Introduction

Diphallia or duplicate penis is an extremely rare embryological anomaly with a wide range of anatomic appearances ranging from small accessory tissues to complete duplications of the phallus, glans, urethras, and bladders, as well as an extensive list of associated abnormalities. The phalluses are usually unequal in size and positionally, can lie side by side, stacked on top of the other in the sagittal plane, or with little association to each other. 1 This paper assesses patients within the literature to summarize diphallia variants, as well as build on the existing classification of diphallia to include a more specific categorization of the phallus soft tissue, the urethral anatomy, and the bladder configurations to create a surgical classification system.

Background

Diphallia is estimated to occur in 1 out of 5–6 million births, with around 100 patients being reported within the literature. The oldest published instance of diphallia was reported in 1609 by Johannes Jacob Wecker; ‘in Bologna during public dissections the cadaver of a man who had a double penis’, however the earliest pictorial record of diphallia dates back to 1862 in the Lupanar (Latin for brothel) in Pompeii, in which a painting on the wall depicted a completely diphallic man (Fig. 1). 2

Fig. 1.

Fig. 1

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Pompeian Lupanar showing Priapus with a double phallus. Image from Galassi et al. accessed January third, 2021.

Embryology

Previously, Cecil 3 submitted four embryological explanations for diphallia. The first includes the bladder, the prostatic urethra, and the penis being derived from a bilateral anlagen, which normally gives a single end product by fusion, meaning diphallia is a product of an incompletely fused anlagen. Next, diphallia may be an atavism, as snakes and lizards normally possess double penis or possibly represents a teratoid structure. Cecil also suggested that it may be a minor degree of duplication, much like supernumery digits, of the individual.

Hollowell et al. however affirm that these explanations are incomplete and suggests that that embryologically, the diphallia anomaly occurs in the fetus between the third and seventh week of gestation, in which an insult hampers normal functioning of the caudal cell mass of the fetal mesoderm at the time of the urogenital sinus separating from the genital tubercle into the penis. Complete diphallia may then stem from longitudinal duplication of the infraumbilical cloacal, with the subsequent mesodermal migration leading to the formation of two separate and complete sets of genital tubercles, genital folds, and genital swellings. 3 , 4

Treatment

Treatment of diphallia is typically approached on an individual basis, as no two patients within the literature are the same. The associated anomalies are a major cause of mortality in diphallia patients, and treatment is usually done in a stepwise manor, with corrections aimed at the anomalies, excision of the more abnormal phallus, excision of the urethra in instances of duplicate urethra, and excision or correction of either duplicate bladders or bladder exstrophy. 1 It may also be difficult to discern which phallus to excise, thus with the proposed classification system, a label can be used to drive the treatment approach, allowing a decision to be made based on the more normal phallus, with the more normal urethral pathway and normal bladder configuration.

Methods

For this paper, a review of published diphallia patients was conducted by one reviewer. Google scholar and PubMed were analysed using key phrases diphallia, double penis, psuedophallia and bifid glans giving a total of 518 articles. Articles were screened based on title and abstract, making sure to include relevant case reports in humans, excluding articles in animals, duplicate articles and non‐English articles. Following the screening of relevant inclusion and exclusion criteria, a total of 76 articles were analysed, which totalled 87 relevant diphallia patients found within the literature. Each patient was then analysed and presented in Table 1. From each patient, the unique anatomical variant for each was then examined. This included first addressing the age of the patient at the time of presentation, followed by categorizing the soft tissue structure of each phallus, the anatomy of the scrotum and testicles, as well as if there was a penoscrotal transposition. The reports were also analysed for their urethral anatomy, and if there was a hypospadias or epispadias present, as well as the bladder configuration. Lastly, the associated abnormalities were summarized based on the reviewers best clinical judgement. Each patient was then classified based on the proposed classification system below.

Table 1.

Literature review of diphallia, associated anatomy and classifications

Author, Year Age at definitive treatment Number of corpora cavernosa Hypospadias/Epispadias Scrotum Testes Penoscrotal Transposition Urethra Bladder Other anomalies Classification
Acimi 5 3 yrs. Phallus 1: 2 Hypospadias Normal Descended bilaterally No Duplicate Single Imperforate anus 1Aα
Phallus 2: 2
Acimi 6 4 mo. Phallus 1: 2Phallus Hypospadias Bifid Descended bilaterally No Duplicate, one functional Single Unilateral Kidney agenesis 1Bα
Lumbosacral meningocele
2: 2
Ureterovesical duplication
Adair and Lewis 7 1 yr. Phallus 1: 2 N/A Normal Descended unilaterally No Single Single Umbilical hernia 7Aα
Unilateral Kidney agenesis
Duplicate Glans
AI‐Herbish and AI‐Samarrai 8 1 mo. Phallus 1: 2 Hypospadias Bifid Descended bilaterally No Duplicate with bifurcation of one urethra Single Unilateral Kidney agenesis 1Αα
Musculoskeletal anomalies
Phallus 2: 2 Hemivertebra and absent first rib
Aihole, 2015 9 1 yr. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Single Pre‐axial polydactyly 7Βα
Solitary Kidney
Duplicate Glans Atrial septal defect
Akgül et al. 10 4 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Duplicate Anal atresia 1Αβ
Duplicate rectum, colon, cecum, appendix and terminal ileum
Phallus 2: 2
Rectovesical fistula
Aleem 11 2 mo. Phallus 1: 1 Epispadias Bifid Descended bilaterally No Single Single Wide diastasis of the pubic bones and partial sacral agenesis. 1Dα
Phallus 2: 1
Ali 12 19 yrs. Phallus 1: 1 N/A Normal Descended bilaterally No Single Single N/A 2Aα
Phallus 2: 2
Arya et al. 13 31 yrs. Phallus 1: 2 Epispadias Bifid Descended bilaterally No Duplicate, no urethral plates Exstrophy N/A 1Dγ
Phallus 2: 2
3 mo. Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Bakheet and Refaei 14 Neonate Phallus 1: 2 N/A Duplicate Duplicate, descended bilaterally No Duplicate Single Duplicate colon, rectum, anus 1Aα
Phallus 2: 2
Bhat et al. 15 1 yr. Phallus 1: 2 Hypospadias Bifid Descended bilaterally No Duplicate, opening normally and into lateral wall of bladder Single N/A 1Cα
Phallus 2: 2
Blanco 16 18 yrs. Phallus 1: 2 Hypospadias Normal Descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Cernach et al. 17 11 mo. Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Duplicate Duplicate anus, one imperforatehemivertebrae and diastasis of pubic symphysis 1Aβ
Phallus 2: 2
Chadha et al. 18 Neonate Phallus 1: 2 N/A Bifid Descended into lateral compartments No Bifurcation at prostatic urethra Single N/A 1Aα
Phallus 2: 2
de Oliveira et al. 19 5 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate, unilateral stenosis Single N/A 1Bα
Phallus 2: 2
Deshpande 20 2 yrs. Phallus 1: 2 N/A Bifid Descended unilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Dewan et al. 21 7 yrs. Phallus 1: 2 N/A Bifid Descended unilaterally No Duplicate, opening normally and into lateral wall of bladder Single Imperforate anus 1Aα
Phallus 2: 2
Djordjevic and Perovic 22 15 mo. Phallus 1: 2 Hypospadias Bifid Descended bilaterally No Duplicate Duplicate Symphyseal diastasis 1Aβ
Phallus 2: 2
Dunn et al. 23 3 yrs. Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Duplicate N/A 1Aβ
Phallus 2: 2
Dutta et al. 24 Neonate Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate, pneumaturia Duplicate Pelvic kidney 1Aβ
Phallus 2: 2 Anorectal atresia
Elsawy et al. 25 1 mo. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate, one hypoplastic Single Inguinal hernia 4Aα
Absent right thumb
Phallus 2: 2
Frollo et al. 26 84 yrs. Phallus 1: 2 Hypospadias Normal Descended bilaterally No No urethra in either phallus Single N/A 1Cα
Phallus 2: 2
Gavali et al. 27 5 yrs. Phallus 1: 2 Hypospadias Normal Descended bilaterally No Single Single Single Kidney agenesis 7Bα
Duplicate Glans
Ghafoori et al. 28 5 yrs. Phallus 1: 1 N/A Normal Descended bilaterally No Duplicate Single N/A 3Aα
Phallus 2: 1
Goad et al. 29 13 yrs. Phallus 1: 2 Hypospadias Normal Descended bilaterally No Single Single N/A 1Aα
Phallus 2: 2
Gupta and Virdi 30 10 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Gyftopoulos et al. 31 Neonate Phallus 1: 2 Hypospadias Bifid Undescended bilaterally No Duplicate Duplicate Horseshoe kidney Partial duplication of distal colonVentricular septum defect 1Aβ
Phallus 2: 2 Hypoplasia of the right leg due to agenesis of the fibula
Cloacal opening at the perineum
Neonate Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate, one with blind ending Duplicate Urethrorectal Y‐fistula 3Aβ
Phallus 2: 2
Hanine et al. 32 8 yrs. Phallus 1: 1 N/A Normal Descended bilaterally No Single Single N/A 2Aα
Phallus 2: 1
Hollowell et al. 13 5 mo. Phallus 1: 2 Hypospadias Bifid Descended bilaterally No Duplicate Duplicate Imperforate anus Perineal fistula 1Aα
Phallus 2: 2 Meningocele
50 yrs. Phallus 1: 2 Hypospadias Bifid Descended into lateral compartments No Duplicate Single Prolapse of the rectum 1Cα
Absence of perineal
Midline infraumbilical musculature
Bilateral inguinal hernias
Phallus 2: 2
Jesus et al. 33 20 yrs. Phallus 1: 2 Hypospadias Normal Descended bilaterally No Duplicate Single N/A 7Cα
Duplicate Glans
Johnson et al. 34 17 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Single N/A 7Aα
Duplicate Glans
Karagöz et al. 35 15 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Single Single N/A 7Aα
Duplicate Glans
Kardasevic et al. 36 Neonate Phallus 1: 2 Hypospadias #NAME? Descended bilaterally No Duplicate Duplicate Atrial and ventricular septal defect 1Aβ
Duplicate right ureter
Phallus 2: 2
Triple spleen
Duplicate right kidney
Karna and Kapur 37 Neonate Phallus 1: 2 N/A Bifid Undescended bilaterally No Duplicate Single Omphalocele 1Aα
Tracheoesophageal fistula
Imperforate anus
Single Kidney agenesis
Duplicate distal ileum, cecum, appendix and colon
Phallus 2: 2
Musculoskeletal anomalies
Kaufman et al. 38 15 yrs. Phallus 1: 2 Hypospadias Normal Descended bilaterally No Duplicate, with third perineal urethra Single Imperforate anus 1Aα
Phallus 2: 2
Keckler 39 Neonate Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Kendrick et al. 40 3 mo. Phallus 1: 2 N/A Normal Descended bilaterally Partial Duplicate, opening normally and into lateral wall of bladder Single N/A 2Bα
Phallus 2: 1
Khorramirouz et al. 41 6 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate, unilateral stenosis Duplicate Horseshoe Kidney 7Bβ
Duplicate Glans
Kırlı et al. 42 Neonate Phallus 1: 2 Epispadias Normal Descended bilaterally No Duplicate Exstrophy Inguinal hernia 1Dγ
Phallus 2: 2
Kundal et al. 43 3 yrs. Phallus 1: 2 Epispadias Normal Descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Landy et al. 44 Neonate Phallus 1: 2 N/A Normal with accessory scrotum Descended bilaterally No Duplicate Single Imperforate anus 1Aα
Phallus 2: 2
Larsen 45 14 yrs. Phallus 1: 1 N/A Normal Descended unilaterally Partial Duplicate Single Atrophic leg 3Aα
Phallus 2: 1
Leite et al. 46 2 yrs. Phallus 1: 1 N/A Normal Descended bilaterally No Duplicate, one with blind ending Single Short oesophagus 3Aα
Phallus 2: 1
Maher et al. 47 Neonate Phallus 1: 2 N/A Duplicate Descended bilaterally No Duplicate Duplicate Separated natal clefts with no anal orifice 1Aβ
Hydronephrotic left kidney
Duplicated colon
Caudal duplication syndrome
Phallus 2: 2
Mandal and Sahi 48 Neonate Phallus 1: 2 N/A Duplicate Duplicate, descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Marti‐Bonmati et al. 49 Neonate Phallus 1: 2 Hypospadias Ectopic scrotal tissue Descended unilaterally No Duplicate, one with blind ending Exstrophy Imperforate anus 1Aγ
Phallus 2: 2
Maruyama et al. 50 Neonate Phallus 1: 1 Hypospadias Bifid Descended unilaterally No Duplicate Single Tracheoesophageal fistula with oesophageal atresia 3Cα
Imperforate anus Bilateral radial limb defects
Cleft palate
Phallus 2: 1 Patent ductus arteriosus, Single umbilical artery
Right megaureterLeft multicystic dysplastic kidney
Matsumoto et al. 51 12 mo. Phallus 1: 1 Hypospadias Bifid Descended bilaterally Complete Duplicate Single Atrial septal defect 2Aα
Phallus 2: 2
Melekos et al. 52 8 yrs. Phallus 1: 1 Hypospadias Normal Descended bilaterally No Duplicate Single Horseshoe Kidney 6Cα
Phallus 2: 1
Mingazzini 53 36 yrs. Phallus 1: 1 N/A Normal Descended bilaterally No Duplicate, unilateral stenosis Single Umbilical hernia 3Aα
Phallus 2: 1
Mirshemirani et al. 54 Neonate Phallus 1: 2 Hypospadias Bifid Descended bilaterally No Duplicate Duplicate Imperforate anus 1Cβ
Phallus 2: 2 Duplicate colon
14 yrs. Phallus 1: 1 Hypospadias Bifid Descended bilaterally No Duplicate Single Imperforate anus 3Cα
Duplicate sigmoid colon
Phallus 2: 1
4 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Duplicate Right inguinal hernia 1Aβ
Phallus 2: 2
12 yrs. Phallus 1: 2 Epispadias Bifid Descended bilaterally No Duplicate Exstrophy Single kidney 4Dγ
Hemi‐Vertebra
Phallus 2: 2
1 yr. Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Duplicate N/A 1Aβ
Phallus 2: 2
9 mo. Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Single Imperforate anus 1Aα
Phallus 2: 2
Mukunda et al. 55 Neonate Phallus 1: 2 N/A Bifid Descended bilaterally No Bifurcation at prostatic urethra Single Meckel's diverticulum 1Eα
Phallus 2: 2
Mutlu et al. 56 9 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Bifurcation at prostatic urethra Single Rotational anomaly of right kidney 2Eα
Phallus 2: 1
Left ureter duplication
Nunez et al. 57 Neonate Phallus 1: 2 Hypospadias Normal Descended bilaterally No Duplicate Single Anorectal malformation 7Cα
Duplicate Glans
Peris 58 17 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Single Single N/A 2Aα
Phallus 2: 0
Priyadarshi 59 1 yr. Phallus 1: 1 Epispadias Bifid Descended bilaterally No Duplicate Single Ectopic bowel segment 3Dα
Phallus 2: 1
Rajarajan 60 23 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Single Anorectal anomalies and colonic duplication 4Cα
Phallus 2: 2
Rao and Chandrasekharam 61 3 mo. Phallus 1: 2 Epispadias Normal Descended bilaterally No Duplicate, one with blind ending Single N/A 7Dα
Duplicate Glans
Remzi 62 14 yrs. Phallus 1: 2 Hypospadias Duplicate Duplicate, descended bilaterally No Duplicate Exstrophy hemivertebrae, lumbosacral scoliosis 1Cγ
Phallus 2: 2
Rock and Gearheart 63 1 yr. Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Duplicate Hypoplastic kidney 1Aβ
Widely separate pubic diastasis
Phallus 2: 2
Sacral insufficiency
Rodriguez 64 Neonate Phallus 1: 2 HypospadiasEpispadias Duplicate Duplicate, descended bilaterally No Duplicate Single (bilobated) Duplicate umbilical cord 1Cα
Phallus 2: 2
Rossete‐Cervantes and Villegas‐Muñoz 65 83 yrs. Phallus 1: 2 N/A Normal Duplicate, descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Savir et al. 66 31 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Single N/A 4Aα
Phallus 2: 2
Sharma et al. 67 Neonate Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate, one with blind ending Single Duplicate colon 1Aα
Phallus 2: 2
Sharma et al. 68 Neonate Phallus 1: 2Phallus N/A Normal Descended bilaterally No Duplicate, one with blind ending Single N/A 1Aα
2: 2
Sina et al. 69 2 mo. Phallus 1: 2 N/A Normal with accessory scrotum Descended bilaterally No Duplicate Single N/A 2Aα
Phallus 2: 1
Smith and Sherer 70 18 yrs. Phallus 1: 2 N/A Normal Descended unilaterally No Duplicate Exstrophy N/A 7Aγ
Duplicate Glans
Solomon et al. 71 Neonate Phallus 1: 2 N/A Normal Descended unilaterally No Duplicate Single Supernumerary kidney 6Aα
Phallus 2: 2
Sotiropoulos et al. 72 12 yrs. Phallus 1: 2 Hypospadias Bifid Descended bilaterally No Duplicate, both blind ending Exstrophy Hypoplastic kidney 1Aγ
Omphalocele
Phallus 2: 2 Bilateral inguinal hernia
Imperforate anus
Colovesical fistula
14 yrs. Phallus 1: 2 Epispadias Bifid Descended bilaterally No Duplicate Exstrophy Agenesis left upper extremity, and a web deformity of the left popliteal region. Rectoperineal fistula 7Cγ
Duplicate Glans
Neonate Phallus 1: 2 N/A Normal Descended bilaterally No Duplicate Exstrophy

Vesicoureteral reflux

Aplastic kidney

 2Dγ
Phallus 2: 2
17 yrs. Phallus 1: 1 N/A Normal Descended bilaterally No Duplicate Duplicate Atrophic right kidney 6Aβ
Phallus 2: 1
Tepeler et al. 73 14 yrs. Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Single N/A 2Aα
Phallus 2: 1
Tirtayasa et al. 74 12 yrs. Phallus 1: 2 Epispadias Bifid Descended bilaterally No Duplicate Single Ectopic bowel segment 1Dα
Phallus 2: 2
Tu et al. 75 Neonate Phallus 1: 2 N/A Bifid Descended bilaterally No Duplicate Single N/A 1Aα
Phallus 2: 2
Vilanova and Raventos 76 30 yrs. Phallus 1: 2 N/A Normal Descended bilaterally No Single Single N/A 2Aα
Phallus 2: 1
Wojewski and Kossowski 77 6 yrs. Phallus 1: 2 N/A Duplicate Duplicate, descended bilaterally No Duplicate Duplicate Talipes equinovarus Ureter stenosis 1Aβ
Phallus 2: 2
Zhang et al. 78 23 yrs. Phallus 1: 2 N/A Normal Duplicate, descended bilaterally No Single Single N/A 7Aα
Duplicate Glans
Zolfaghari et al. 79 4 mo. Phallus 1: 2 Hypospadias Normal Descended bilaterally No Duplicate, one with blind ending Single Bilateral congenital hip dislocation, severe right talipes equinovarus and hypotrophy of the right lower limb. Inguinal hernia 1Aα
Phallus 2: 2
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Classification

Schneider 80 has previously classified diphallia into four main categories:

  1. Duplication of the glans alone

  2. Bifid diphallia

  3. Complete diphallia with each penis having two corpora cavernosa and a corpus spongiosum

  4. Pseudodiphalia in which there is a rudimentary accessory atrophic penis existing independently of the normal penis

Our proposed classification is based on anatomical variants found within the literature on diphallia, which builds on Schneider's classification in order to provide a more specific description of the phallus soft tissue, as well as include a description of the pathway of the most normal urethra and the bladder configuration. The classification method has been proposed to be used to simplify the categorization of diphallia patients, using the system to classify the most intact phallus, the most normal urethra, and whether there is an additional surgical step regarding the bladder. The system will place the anatomy of diphallia into specific categories based on the structure of the phallus, the urethral anatomy and the bladder formations. Table 2 below displays the proposed categories.

Table 2.

Proposed classifications of phallus, urethra and bladder found in Diphallia

Phallus
Character Type
Separate phalluses, 3 corpora each 1
Separate phalluses, 3 corpora in only one 2
Separate phalluses, neither contain 3 corpora 3
Phalluses contained within same shaft skin, 3 corpora each 4
Phalluses contained within same shaft skin, 3 corpora in only one 5
Phalluses contained within same shaft skin, neither contain 3 corpora 6
Bifid glans 7
Urethra to most normal phallus
Character Type
Normal urethra present A
Urethral stenosis B
Hypospadias C
Epispadias D
Bifurcation E
Bladder
Character Type
Single α
Double β
Exstrophy γ
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Results

With a review of the literature, and application of the proposed classification system, common diphallia configurations can be displayed. While these individuals may still differ in terms of specific anatomy, such as the relation of the phalluses to one another, the specific pathway of the urethras or the associated anomalies, the classification and the frequency in which they occur is based on the soft tissue make‐up of the phalluses, the urethra as it pertains to the most normal urethra and the bladder configuration. Of the reviewed literature with sufficient information, our classification showed that diphallia patients categorized as 1Aα contributed to the largest percentage of reports at 26.4%. The next most frequent pattern seen was 1Aβ, where the soft tissue and urethra were structurally normal, however there was a duplicate bladder, which made up 12.6% of the patients reviewed. The 2Aα configuration was the next most seen classification within the literature, appearing in 7% or 8.0%, followed by configurations 1Cα, 3Aα, 7Aα, which each contributed to 4.6% of the patients. The remaining reports and the corresponding classifications can be reviewed in Table 3. Table 4 then depicts the most common configurations found within the literature and additional examples of diphallia, which shows a graphic of the corresponding anatomy, an example of the gross anatomy extracted from their corresponding reference and the patient that correspond to the detailed classification.

Table 3.

Proportion of diphallia anatomical variants within the literature

Classification Count Percent (%)
1Aα 23 26.4
1Αβ 11 12.6
2Aα 7 8.0
1Cα 4 4.6
3Aα 4 4.6
7Aα 4 4.6
1Aγ 2 2.3
1Dα 2 2.3
1Dγ 2 2.3
3Cα 2 2.3
4Aα 2 2.3
7Bα 2 2.3
7Cα 2 2.3
1Bα 2 2.3
1Cβ 1 1.1
1Cγ 1 1.1
1Eα 1 1.1
2Bα 1 1.1
2Dγ 1 1.1
2Eα 1 1.1
3Aβ 1 1.1
3Dα 1 1.1
4Cα 1 1.1
4Dγ 1 1.1
6Aα 1 1.1
6Aβ 1 1.1
6Cα 1 1.1
7Aα 1 1.1
7Aγ 1 1.1
7Bβ 1 1.1
7Cγ 1 1.1
7Dα 1 1.1
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Table 4.

Most common anatomical varriants for diphallia

Classification Diagram Gross anatomy References
1Aα graphic file with name ANS-92-2053-g004.jpg graphic file with name ANS-92-2053-g008.jpg Chadha et al. 18
1Αβ graphic file with name ANS-92-2053-g009.jpg graphic file with name ANS-92-2053-g011.jpg Maher et al. 47
2Aα graphic file with name ANS-92-2053-g001.jpg graphic file with name ANS-92-2053-g013.jpg Sina et al. 69
1Cα graphic file with name ANS-92-2053-g010.jpg graphic file with name ANS-92-2053-g003.jpg Bhat et al. 15
3Aα graphic file with name ANS-92-2053-g007.jpg graphic file with name ANS-92-2053-g002.jpg Leite et al. 46
7Aα graphic file with name ANS-92-2053-g012.jpg graphic file with name ANS-92-2053-g014.jpg Zhang et al. 78
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Associated anomalies

Upon review of the literature, several diphallia patients present with no other associated anomalies. However, several abnormalities can be seen in other diphallia patients. These anomalies arise from both genitourinary and gastrointestinal systems, as well as some reports seeing musculoskeletal or cardiovascular anomalies. The majority of the malformations fall within the former two. Genitourinary abnormalities include the presence of either a duplicate bladder 10 , 13 , 17 , 22 , 23 , 24 , 31 , 36 , 41 , 47 , 54 , 63 , 72 or bladder exstrophy, 13 , 42 , 49 , 62 , 70 , 72 , 77 as well as duplicate ureters, 36 , 56 vesicoureteral reflux, 47 , 72 and ureteric stenosis. 77 Further anomalies within the urinary system include issues and malformations pertaining to the kidneys, which include single kidney agenesis, 7 , 8 , 9 , 37 , 54 , 63 , 72 duplicate unilateral kidney, 36 pelvic kidney 24 and horseshoe kidney. 31 , 41 , 52 Fistulas pertaining to the urinary system were also a common association with rectovesical and urethrorectal 10 , 31 , 72 being noted. Next, there is also a wide range of gastrointestinal abnormalities that appear to arise in patients with diphallia. The most seen is the presence of an imperforate anus. 5 , 13 , 17 , 24 , 37 , 38 , 44 , 47 , 49 , 50 , 54 , 72 There can also be further duplication of various aspects of the gastrointestinal system associated with diphallia, which includes duplication of the rectum, colon, cecum, appendix, and terminal ileum. 10 , 14 , 31 , 37 , 47 , 54 , 60 , 67 Along with the mentioned fistulas mentioned above, perineal 13 and tracheoesophageal fistula 37 , 50 have been reported as associated abnormalities pertaining to the gastrointestinal tract. It was also noted that there were hernias associated with diphallia patients, mostly those occurring at the umbilicus, 7 , 25 , 41 , 53 however inguinal hernias 54 , 72 , 79 are also cited within the literature. Further gastrointestinal anomalies then include ectopic bowel segments, 59 , 74 omphalocele 37 , 72 and Meckel diverticulum. 55

In terms of musculoskeletal and cardiovascular abnormalities, there is a wide range of malformations shown in the literature that are associated with diphallia. Two common associations include both hemivertebra 8 , 54 , 62 and a wide diastasis of the pubic bone 11 , 17 , 22 , 63 with further musculoskeletal malformations comprising of menigocele 6 , 13 and talipes equinovarus 77 , 79 and less commonly pre‐axial polydactyly, 9 partial sacral agenesis, 11 agenesis and hypotrophy of digits or limbs, 25 , 31 , 45 , 72 , 79 or bilateral congenital hip dislocations. 79 Cardiovascular malformations are more rare, however present in multiple patients, which include abnormalities such as atrial septal defects. 9 , 51 A summary of associated abnormalities pertaining to specific patients is outlined in Table 1.

Table 5 then displays the most commonly seen abnormalities seen in concordance with diphallia. Most commonly, an imperforate anus was seen associated with diphallia patients which was shown in 13.8% of the reviewed patients. Next, 9.2% of the patients also saw further duplication along the gastrointestinal tract, with duplications of either the rectum, colon, cecum, appendix or terminal ileum being reported. Single kidney agenesis and inguinal hernias were then the next most seen abnormalities, arising in 8.0% and 6.9% of the patients, respectively. Lastly, the most common musculoskeletal anomaly associated with diphallia was limb agenesis or hypotrophy, which appeared in 5.7% of the diphallia cases.

Table 5.

Proportion of associated abnormalities

Associated Abnormality Count Percent (%)
Gastrointestinal
Imperforate anus 12 13.8
GIT duplication 8 9.2
Anorectal malformation 5 5.7
Ectopic bowel segments 2 2.3
Omphalocele 2 2.3
Oesophageal atresia with tracheoesophageal fistula 2 2.3
Meckel diverticulum 1 1.1
Genitourinary
Single renal agenesis 7 8.0
Horseshoe kidney 3 3.4
Duplicate ureters 2 2.3
Vesicoureteral reflux 2 2.3
Pelvic kidney 1 1.1
Duplicate kidney 1 1.1
Ureteric stenosis 1 1.1
Muskuloskeletal
Limb agenesis/hypotrophy 5 5.7
Wide diastasis of pubic bone 4 4.6
Hemivertebra 3 3.4
Meningocele 2 2.3
Talipes equinovarus 2 2.3
Sacral agenesis 1 1.1
Bilateral hip dislocations 1 1.1
Cardiovascular
Atrial septal defect 2 2.3
Hernias
Inguinal 6 6.9
Umbilical 2 2.3
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Discussion

The paper highlights an overview of diphallia, illustrating different aspects of the anomaly including history, embryology and treatments. It also expands to provide a classification system that is built on previous works to give a system in which future patients can be categorized and compared. The results then feature the most common anatomical variations, showing that roughly 25% of the published instances have two phalluses with 3 corpora, at least a single normal urethra and a single bladder. The literature review and the published articles however may be biased as typically the more unique and interesting patients are presented, leaving a potentially large number of unpublished reports that could contribute to the current review and proposed classification system. The results regarding associated anomalies potentially hold a similar bias, in that unique abnormalities may have been focused on, leaving out seemingly minute associations. Lastly, as new patients may be presented, and different variants may arise in which the proposed classification system may or may not encompass, or penile anomalies such as triphallia or triple penis, as reported by Jabali et al. 81 get reported, the current proposed classification may need to be modified.

Conclusion

Following a literature review, each diphallia patient is a unique variant with its own anatomical configuration and associated anomalies. This has led to the proposed classification system that builds on previous bodies of work to categorize each patient based on the most normal aspects of the diphallia. By classifying the structure of the phallus that is to be kept, the pathway of the urethra present within the most normal phallus and the bladder morphology, a surgical approach can be broached and executed to ensure a satisfactory functional goal, with preserved continence, erectile function, and cosmetic outcomes.

Conflict of interest

The review is not registered, and the protocol was not prepared. Support from the Paediatric Surgery and Urology Department at the Queensland Children's Hospital. None declared.

Author contributions

Dylan John Kendrick: Conceptualization; data curation; formal analysis; investigation; methodology; visualization; writing – original draft; writing – review and editing. Roy Mark Kimble: Project administration; supervision; writing – review and editing.

D. J. Kendrick BSc, MSc; R. M. Kimble MBCHB, FRACS, FRCSP, MD.

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