Ano-Scrotal Distance (ASD) : Is It A Marker For The Severity of Chordee?

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Journal of Pediatric Urology (2021) 17, 670.e1e670.

e5

Ano-Scrotal Distance (ASD) : Is it a


marker for the severity of chordee?

Johannes Wirmer, Michael Sennert, Ahmed T. Hadidi *


Hypospadias Centre,
Department of Pediatric Summary Results
Surgery, Emma and Sana 817 boys were included. The patients were classified
Klinikum Offenbach Hospitals, Objective according to the hypospadias grades I (180 pt), II
Germany
To evaluate the relation between ASD and the (415 pt), III (120 pt) and IV (102 pt). The median ASD
severity of chordee and hypospadias in a referral for group I was 3.07 cm (range 1.0e5.5), for group II
* Correspondence to: Ahmed T center. 3.10 cm (range 1.5-5.0), for group III 2.40 cm (range
Hadidi, Max-Planck Str. 2,
1.5e54.5) and for group IV 2.10 cm (range 1.0e3.5).
Seligenstadt, D - 63500,
Germany, Tel.: þ49 174 205
Methods The average ASD of the patients in Grade III and
6905; fax: þ49 6182 843 0293 The distance between the anal verge and proximal IV (2.26 cm) was significantly shorter than the
Prof.Hadidi@hypospadiezen- scrotal edge (ASD) was measured in hypospadias pa- average ASD of the patients in Grade I and II
trum.de, ahmedthadidi@ tients between 6 and 18 months between 2016 and (3.09 cm) (p < 0.05). 184 patients had an ASD
yahoo.de (A.T. Hadidi) 2020 in the operating room under anaesthesia. Pa- 2.0 cm. 38% of patients with glanular hypospadias
tients older than 36 months and younger than 6 (grade I) with ASD  2.0 cm had severe chordee.
Keywords months were excluded. The patients were classified
Penis; Ano-scrotal distance according to meatus into: Grade I (glandular or chor-
urologic surgical procedures;
dee without hypospadias), II (distal), III (proximal) Conclusion
Chordee; Hypospadias; Ure-
thra; Male
and IV (perineal). Chordee was assessed by erection This study shows that short ASD is a helpful marker
test and classified into 2 groups; Group I: no chordee of the severity of chordee and hypospadias. Patients
or mild chordee <30 and Group II: with severe chor- with glanular hypospadias and short ASD have 38%
Received 15 January 2021
Revised 16 April 2021
dee >30. Short ASD was defined as  2.0 cm. chance of having severe chordee.
Accepted 4 June 2021
Available online 12 June 2021

https://doi.org/10.1016/j.jpurol.2021.06.010
1477-5131/ª 2021 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.

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Ano-Scrotal Distance 670.e2

Introduction the severity of chordee. However, it is difficult to assess the


severity of chordee during the outpatient examination
The standard classifications of hypospadias according to the without erection especially when the baby is crying and
meatus location do not consider the severity of chordee. moving. While it is expected to encounter severe chordee
The severity of chordee influences the surgical technique in severe forms of hypospadias, it is also encountered in
and the incidence of complications. some mild forms, leading to unpleasant surprises during
The Ano-Genital and Ano-Scrotal Distance (AGD and ASD) surgery.
has been identified as a marker for androgen exposure in Recent studies suggest an association of hypospadias in
utero in numerous animal studies, with a short distance general with short AGD and ASD [9, 17, 18]. However, the
reflecting decreased androgen exposure [1]. A study ASD in patients with hypospadias has so far only been
examining the ASD in 371 male newborns found an average examined on a small patient series [9]. An association be-
ASD of 2.47 mm [2]. tween the ASD and severity of chordee has not been
Hypospadias on the other hand, is second commonest reported.
congenital malformations in boys after undescended testis This study was designed to assess the relationship of ASD
with a rising prevalence [3e5]. However, many coherences with the severity of chordee and the severity of hypospa-
in this common congenital anomaly remain unclear [6e8]. dias in a large referral center.
These clinical classifications lack international stan-
dardization [6, 9]. However, the classification of hypospa- Methods
dias into four grades (glanular, distal, proximal and
perineal) seems to become more popular [10]. Meanwhile, In a prospective study, boys between 6 and 18 months un-
more than 300 surgical techniques have been described for dergoing hypospadias surgery in the center between
correction [10]. In our center, we use 4 surgical techniques January 2016 and December 2020 were included in the
for the different grades of hypospadias [10e16]. study. Patients older than 36 months or younger than 6
To choose the appropriate surgical technique, the sur- months were excluded. The study obtained the approval of
geon needs to identify the grade of hypospadias as well as the institutional ethics committee.

Figure 1 Ano-Scrotal Distance (ASD). a. glanular hypospadias with 5 cm ASD. b. Distal hypospadias with 3 cm ASD. c. Mid-penile
hypospadias with the ASD Z 3 cm. Notice that 3 cm is about 2 finger breadth. d. Proximal hypospadias with 2 cm ASD. e. Perineal
hypospadias with 1.5 cm ASD. Observe in (c) that 3 cm is about 2 fingers ‘breadth. This is helpful to have a rough estimation of ASD
in the outpatient clinic especially if the child is moving.

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670.e3 J. Wirmer et al.

Figure 2 A 12 months old child with glanular hypospadias and severe chordee. a. Examination under anaesthesia showed that the
penis was curved and the ASD was short (1 cm). b. Close up showing that ASD was 1 cm. c. Close up showing the glanular hypo-
spadias. d. Notice the extensive dissection including excision of the outer Longitudinal layer of Tunica Albuginea around the
urethra. However, the urethra was not completely mobilized to maintain its blood supply. Also observe how the sub-coronal incision
retracted down from the sub-coronal level to the proximal penis. e. Erection test confirmed that the penis was straight. f. The final
appearance after glans and skin closure.

Patients were classified according to the location of the no chordee or mild chordee, less than 30 corrected by
meatus into 4 grades: Grade I (glanular or chordee without degloving and severe chordee (more than 30 ) requiring
hypospadias), Grade II (distal to the mid penis), Grade III additional procedures for chordee correction other than
(proximal, from mid penis to penoscrotal junction) and degloving. These include division of the urethral plate,
Grade IV (proximal to penoscrotal junction). excision of the outer layer of tunica albuginea and (when
The Ano-Scrotal Distance (ASD) was measured amongst indicated) incision of the inner layer of tunica albuginea
other anatomical landmarks under general anaesthesia. and placing a flap or graft. The mean ASD and the per-
Anoscrotal distance was defined as the distance between centage of severe chordee for every hypospadias grade
the anterior anal verge and proximal edge of the scrotum were documented.
where the perineal skin changes from smooth to corrugated Furthermore, the average ASD of patients with a mild
as described by Sathyanarayana et al. in the infant devel- form of hypospadias (Grade I and II) was compared to the
opment and the environment study (TIDES) [2] and average ASD of patients with more severe form of hypo-
measured with a flexible ruler by midline or symmetry spadias (Grade III and IV) and a two-sample t-test with
(Fig. 1). assumed unequal variance was used to determine signifi-
Short ASD was defined as equal or smaller than 2.0 cm cance of difference along the two unpaired groups.
and the severity of chordee was assessed intra-operatively. Patients with grade I hypospadias and severe chordee
Chordee was assessed under anaesthesia by erection test required more aggressive approach than simple degloving
(Fig. 2) using a goniometer and was classified into 2 groups: (Fig. 2). This may include aggressive excision of fascia,

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Ano-Scrotal Distance 670.e4

Table 1 ASD and severe chordee to severity of hypospadias total number of patients Z 817 patients.
Grade I II III IV
Total 180 415 120 102
Mean ASD (cm) 3.07 3.10 2.40 2.10
Range(cm) 1.0e5.5 1.5e5.0 1.0e4.5 1.0e3.5
No. Pt. with ASD  2,0 cm 21 51 50 62
% Pt. with ASD  2,0 cm 11.67% 12.29% 41.67% 60.78%
No. Pt. with severe chordee 24 9 25 102
% of Pt. severe chordee 13.33% 2.17% 20.83% 100%

division of the tunica albuginea longitudinally as it bi- As the TIDES study described a mean ASD of 2.47 cm in
furcates to enclose the urethra and excision of the outer 371 male newborns [2], we defined a short ASD as equal or
layer of tunica albuginea deep to the urethra. In 3 patients smaller than 2.0 cm, considering the expected increase of
with chordee without hypospadias, the authors had to the ASD during the first 3 month of birth.
incise the tunica albuginea and put dermal graft. The results of the study on 817 patients with hypospa-
dias showed a direct relation of short ASD with the severity
of hypospadias. The majority (95%) of patients with distal
Results and proximal (Grade II & III) hypospadias had a normal ASD.
Cox et (2017) demonstrated that, patients with hypospadias
The grade of hypospadias and ASD measurements were in general have a shorter ASD compared to a healthy pop-
documented in 817 Patients between 6 and 36 months. ulation in a small sample size without differentiation of the
There were 180 cases of hypospadias Grade I, 415 Grade II, hypospadias grade [9].
120 Grade III and 102 Grade IV. To our knowledge, this is probably the first report in
The median ASD overall was 2.86 cm. The median ASD for literature that shows the relation between short ASD and
group I was 3.07 cm (range 1.0e5.5), for group II 3.10 cm the severity of chordee.
(range 1.5-5.0), for group III 2.40 cm (range 1.5e4.5) and for This is perhaps of particular importance in patients with
group IV 2.10 cm (range 1.0e3.5) (Table 1). The average ASD glanular hypospadias or chordee without hypospadias. Ac-
of the patients in group I and II (3.09 cm) was significantly cording to the study, if the surgeon finds that the ASD is less
longer than the average ASD of the patients in group III and IV than 2 finger width (2 cm), there is a 38% chance that the
(2.26 cm) (p < 0.05) (Table 2). child has severe chordee not correctable by simple
45% of the 184 Patients with short ASD (2.0 cm) had degloving. This is of great importance when informing the
severe chordee (see Table 2). All patients with perineal parents of success rate, possible complications and perhaps
hypospadias and short ASD had severe chordee (>30 ) that hospital stay.
necessitated division of the urethral plate. Interestingly, Management of Grade I or glanular hypospadias may not
more than 1/3 of patients (38%) in Grade I had severe as easy as one may expect with mild hypospadias especially
chordee that could not be corrected by simple degloving. if there is associated severe chordee. This may be sus-
This is different from patients with Grade II or III Hypo- pected in the outpatient clinic if the ASD is shorter than
spadias with short ASD (only 5% had severe chordee) 2 cm as was the case with this child. This is of importance
(Table 3). for the less experienced surgeon to choose the simple cases
and refer cases with severe chordee to a more specialized
center.
Discussion One limitation of the study is that the measurement can
be operator dependent. However, the 3 authors have
noticed that difference of measurement is about 2e3 mm
In this study, the ASD was measured in 817 children with
among them. The second limitation is that using a flexible
hypospadias between 6 and 36 months. We excluded all
ruler does not provided 100% accurate measurements.
younger and older children, since other studies have shown
that ASD and Ano-Genital Distance (AGD) are expected to
increase rapidly during the Mini Puberty Period (first 6
months) and plateau after one year of age [19]. Table 3 Incidence of short ASD (2.0 cm) and severe
Chordee total Z 184.
Table 2 The mean ASD group I þ II vs group III þ IV Grade I II III IV
total Z 817.
No. of patients with 21 51 50 62
Grade I-II III-IV ASD 2.0 cm
Total 595 222 No. of patients with 8 4 8 62
Mean ASD (cm) 3.09 2.26 severe chordee
Range(cm) 1.0e5.5 1.0e4.5 Percentage of patients 38% 4.8% 4.91% 100%
p < 0.05 with severe chordee

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670.e5 J. Wirmer et al.

It must be pointed out that some patients with severe [6] Springer A, van den Heijkant M, Baumann S. Worldwide
forms of hypospadias and severe chordee may still have prevalence of hypospadias. J Pediatr Urol 2016. https:
normal ASD. However, all patients with perineal hypospa- //doi.org/10.1016/j.jpurol.2015.12.002.
dias and short ASD had severe chordee. [7] Chen MJ, Karaviti LP, Roth DR, Schlomer BJ. Birth prevalence
of hypospadias and hypospadias risk factors in newborn males
in the United States from 1997 to 2012. J Pediatr Urol 2018.
Conclusion https://doi.org/10.1016/j.jpurol.2018.08.024.
[8] van der Zanden LFM, van Rooij IALM, Feitz WFJ, Franke B,
The study suggests that ASD is a helpful marker to identify Knoers NVAM, Roeleveld N. Aetiology of hypospadias: a sys-
the severity of hypospadias and the severity of chordee. tematic review of genes and environment. Hum Reprod Up-
date 2012. https://doi.org/10.1093/humupd/dms002.
This may help the surgeon to identify patients with severe
[9] Cox K, Kyriakou A, Amjad B, O’Toole S, Flett ME, Welsh M,
chordee, plan the appropriate technique and to give the et al. Shorter anogenital and anoscrotal distances correlate
parents a more reliable information during outpatient with the severity of hypospadias: a prospective study. J
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Conflict of interest [10] Hadidi AT. History of hypospadias: lost in translation. J Pediatr
Surg 2017;52. https://doi.org/10.1016/j.jpedsurg.2016.11.
004.
None. [11] Hadidi AT. Perineal hypospadias: back to the future chordee
excision & distal urethroplasty. J Pediatr Urol 2018. https:
Funding //doi.org/10.1016/j.jpurol.2018.08.014.
[12] Hadidi AT. Perineal hypospadias; the bilateral based (BILAB)
skin flap technique. J Pediatr Surg 2014. https://doi.org/10.
This research did not receive any specific grant from 1016/j.jpedsurg.2013.09.067.
funding agencies in the public, commercial, nor not-for- [13] Hadidi AT. Proximal hypospadias with small flat glans: the
profit sectors. lateral-based onlay flap technique. J Pediatr Surg 2012. https:
//doi.org/10.1016/j.jpedsurg.2012.06.027.
[14] Hadidi AT. The slit-like adjusted Mathieu technique for distal
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