Download as pdf or txt
Download as pdf or txt
You are on page 1of 6

+ MODEL

Journal of Pediatric Urology (2013) xx, 1e6

The GMS hypospadias score: Assessment of inter-


observer reliability and correlation with post-
operative complications
Laura S. Merriman, Angela M. Arlen, Bruce H. Broecker, Edwin A. Smith,
Andrew J. Kirsch, James M. Elmore*

Department of Urology, Children’s Healthcare of Atlanta, Emory University School of Medicine, Georgia Pediatric Urology,
Atlanta, GA, USA

Received 22 January 2013; accepted 8 April 2013

KEYWORDS Abstract Objective: An agreed upon method for describing the severity of hypospadias has
Hypospadias; not been established. Herein we assess the inter-observer reliability of the GMS hypospadias
Chordee; score and correlate it with the risk of a post-operative complication.
Score; Methods: A 3-component method for grading the severity of hypospadias was developed
Grade; (GMS). Eighty-five consecutive patients presenting for hypospadias repair were graded inde-
Complications; pendently by at least 2 surgeons using the GMS criteria. Scores were compared statistically
Children to determine agreement between the observers. The outcomes of these patients were then
reviewed to determine how the GMS score correlates to the risk of a surgical complication.
Results: The G, M, and S scores had excellent agreement between observers. The GMS total
score was exactly the same or differed by one point in 79/85 (93%) of patients. The complica-
tion rate was 5.6% for patients with a GMS score of 6 or less, but was 25.0% for patients with a
GMS score greater than 6.
Conclusions: The GMS score provides a concise method for describing the severity of hypospa-
dias and appears to have high inter-observer reliability. The GMS score also appears to corre-
late with the risk of a surgical complication.
ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.

Introduction exists along a continuum. Some cases are only a minor


cosmetic concern, while other cases may be severe and
Hypospadias is one of the most common congenital anom- effect urinary and/or reproductive function. Given this
alies in boys. The severity of the anomaly varies widely and widely variable presentation, as well as anatomical nuances

* Corresponding author. Emory University School of Medicine, Georgia Pediatric Urology, 5545 Meridian Mark Rd, Suite 420, Atlanta, GA
303042, USA. Tel.: þ1 404 252 5206; fax: þ1 404 252 8604.
E-mail address: jmelmoremd@gmail.com (J.M. Elmore).

1477-5131/$36 ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.jpurol.2013.04.006

Please cite this article in press as: Merriman LS, et al., The GMS hypospadias score: Assessment of inter-observer reliability and corre-
lation with post-operative complications, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/j.jpurol.2013.04.006
+ MODEL
2 L.S. Merriman et al.

that make every case unique, hypospadias is difficult to fashion. The G score was based on assessment of the glans
describe in a concise and standardized manner. and urethral plate. In cases where there was a discrepancy
The position of the urethral meatus is the most common between glans size and urethral plate quality, the higher
way to classify the severity of hypospadias, however meatal score was used. The S score was assessed in a standardized
position is only one of several possible anatomic factors fashion by gently pushing downward on the peno-pubic
that effect outcomes. Other features of the anomaly, such junction from above while simultaneously gently pushing
as the quality of the urethral plate, size of the glans, and downward on the peno-scrotal junction from below. No
the severity of chordee, may impact the success of surgery rulers or other measuring devices were utilized. The highest
as much as meatal location. These factors are not typically M score was 4 and included scrotal and perineal hypospa-
or precisely reported in most outcomes studies. Hence, a dias if encountered.
confounding problem of many hypospadias outcomes In total, 7 attending pediatric urologists participated in
studies is a common one e how is “mild”, “moderate” or this study along with 2 pediatric urology fellows and 4
“severe” defined? urology residents. New surgeon scorers were oriented to
In an effort to address the need for standardized criteria the GMS scale by a review of the criteria. The standardized
to classify the severity of hypospadias, the GMS hypospa- method for simulating an erection to assess chordee was
dias scale was developed (Fig. 1). This scale was developed also described. Aside from this no special training, photo-
as a means to qualitatively score the severity of hypospa- graphs, or demonstrations were utilized. After patients
dias based on easily observable features of the glans (G), were scored, the surgeons were permitted to discuss the
meatus (M), and penile shaft (S). Each of the three com- scores and any discrepancies. Verbal and non-verbal cues,
ponents is scored numerically on a scale of 1e4 with more however, were prohibited during scoring and no scores
unfavorable characteristics being assigned higher values. could be changed once finalized. Patients who had surgery
These values are then summed to determine the GMS total at our ambulatory surgery centers were not included in this
score. The lowest possible GMS score, therefore, is 3 (very study since more than one surgeon was not available for
mild hypospadias) and the highest score is 12 (severe scoring. Individual G, M, and S scores as well as total GMS
hypospadias). Representative photographs of patients scores were then compared statistically to determine
categorized using the GMS criteria are shown in Fig. 2. Note agreement between the raters.
that certain qualities are difficult to demonstrate photo- Statistical analyses were performed using SAS 9.2 (Cary,
graphically and that there is some subjectivity to the scale. NC). Statistical significance was assessed using a 0.05 level
It is also important to point out that the G score is used to of statistical significance. Descriptive statistics (e.g.,
assess both glans size and the quality of the urethral plate. means and standard deviations or frequencies and per-
In Fig. 2, the photographs shown were taken to emphasize centages) were calculated for each component of the GMS
differences in urethral plate quality and not necessarily score as well as the total score. The Wilcoxon rank sum test
glans size. was used to compare median GMS scores (and each
The purpose of the current study is two-fold: 1) to component score) to assess whether differences existed
determine if the GMS score is reproducible between ob- between the patients being scored; specifically, for patient
servers and 2) to determine if and how the GMS score cor- differences related to the severity of the condition.
relates to the risk of a surgical complication. Agreement between raters was assessed using the Intra-
class Correlation Coefficient (ICC).
A chart review of those patients scored by GMS criteria
Materials and methods was then conducted 6 months following repair and perti-
nent data was collected. The data collected included the
Following IRB approval, a group of consecutive patients GMS component and total scores, age, type of repair, and
having primary hypospadias repair at our inpatient hospitals any surgical complication. The GMS score was then corre-
were scored using the GMS criteria. Patients were assessed lated to the risk of a post surgical complication.
while under anesthesia and immediately prior to any sur-
gical manipulation by at least 2 surgeons in a blinded
Results
Glans (G) Score:
1. Glans good size; healthy urethral plate, deeply grooved Eighty-five patients with a mean age of 8.8 months (range 3
2. Glans adequate size; adequate urethral plate, grooved monthse8.6 years) were scored using the GMS method. The
3. Glans small in size; urethral plate narrow, some fibrosis or flat distribution of scores is shown in Fig. 3. The average total
4. Glans very small; urethral plate indistinct, very narrow or flat
GMS score was 6.2 (range 3e12) and the average G, M, and S
Meatus (M) Score: scores were 2.0, 2.1, and 2.1 respectively. The rating sur-
1. Glanular
2. Coronal Sulcus geons had complete agreement on component scores 78%,
3. Mid or Distal Shaft 79%, and 85% of the time for the G, M, and S scores
4. Proximal shaft, penoscrotal respectively. In only 1 case did a component score differ by
Shaft (S) Score: more than one (Table 1). The GMS total score was the same
1. No chordee in 50 of the 85 (58.8%) cases and differed by 1 in another 29
2. Mild (< 30°) chordee
3. Moderate (30 - 60°) chordee
(34.1%). Therefore, the total GMS score was the same or
4. Severe (> 60°) chordee differed by one in 79 of 85 (93.0%) cases. Statistical analysis
showed the G, M, and S scores to have very good to
Figure 1 GMS scoring criteria. excellent agreement between observers. The ICC ranged

Please cite this article in press as: Merriman LS, et al., The GMS hypospadias score: Assessment of inter-observer reliability and corre-
lation with post-operative complications, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/j.jpurol.2013.04.006
+ MODEL
The GMS hypospadias score 3

Figure 2 Representative photographs of the GMS scoring criteria. Note that for the G scores, the examples shown are to
emphasize differences in urethral plate quality rather than glans size.

from 0.82 to 0.90. There was no statistically significant attending-fellow group was noted. Eighteen of the patients
change or improvement in agreement over time for any of were also scored by two attending pediatric urologists. A
the unique observer pairs or over the study period. trend toward better agreement was noted, however this
For statistical purposes, the two observers compared was not found to be statistically significant.
were the primary surgeon and the pediatric urology fellow The surgical technique chosen for hypospadias repair
in 27 of 85 (32%) cases or an upper level urology resident in was left up to the attending surgeon. In general TIP or
58 of 85 cases (68%). There were no statistical differences Thiersch Duplay repairs were used for mild to severe
in agreement between the pediatric urology fellow and hypospadias. The urethral plate was incised at the sur-
resident but a trend toward matched agreement for the geon’s discretion to allow for tension-free tubularization.
GAP and MAGPI repairs were utilized for glanular hypospa-
dias. Hypospadias repair types included: TIP in 51 (60%),
Thiersch-Duplay in 19 (22%), GAP in 6 (7%), MAGPI in 4 (5%),
a 2-stage repair in 4 (5%) and 1 patient had a flip-flap repair.
Eleven patients (13%) had a post-operative complication,

Table 1 Frequency of agreement between raters and


measures of inter-rater reliability.a
Variable Complete Differ by 1 Differ by 2 ICC
agreement
G 66 (78%) 18 (21%) 1 (1) 0.82
M 67 (79%) 18 (21%) 0 (0) 0.84
S 72 (85%) 13 (15%) 0 (0) 0.90
a
All measures of agreement were statistically significant
Figure 3 Distribution of G, M, and S scores of the study
(p < 0.05).
cohort.

Please cite this article in press as: Merriman LS, et al., The GMS hypospadias score: Assessment of inter-observer reliability and corre-
lation with post-operative complications, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/j.jpurol.2013.04.006
+ MODEL
4 L.S. Merriman et al.

including glans dehiscence in 2, urethral fistula in 7, and


Table 2 Average G, M, S and GMS total scores for patients
meatal stenosis in 3. One patient developed both meatal
who developed a complication, categorized by complication
stenosis and a urethral fistula.
type.
The complication rate was 5.6% (3/53) for patients with
a GMS score of 6 or less versus 25.0% (8/32) for patients Complication type G M S GMS total
with a GMS score of greater than 6. Fig. 4 shows the dis- Fistula (n Z 7) 2.1 2.4 2.1 6.6
tribution of GMS scores for the study cohort as well as the Meatal stenosis (n Z 3) 3.0 3.3 2.7 9.0
number with each score who developed a complication. Dehiscence (n Z 2) 3.5 2.0 3.5 9.0
The average total GMS score for those patients with post-
operative complications was 7.7 (range 5e12, median 7.0)
and differed significantly from the average total GMS score lacking and highlight the difficulties encountered when
of 5.9 (range 3e12, median 5.0) for those patients who did trying to compare one study to another without a stan-
not have a complication (p Z 0.017, 95% CI 3.08 to dardized classification scheme. Giannantoni, in a recent
0.32). The average component and total GMS scores for editorial regarding hypospadias, also comments on the
those patients who developed a complication, according to need for use of a “same language” and points out the need
complication type, are shown in Table 2. There was no for a better definition of moderate and severe with regards
statistical difference in complication rate based on repair to chordee [6]. Indeed, inconsistency with descriptive
type, but 2 of 4 patients requiring a 2-stage repair devel- terms for hypospadias and the lack of a standardized
oped a complication. method to assess severity of the entire complex limits the
comparison of outcomes studies. Indirectly, this may
impede our ability as a specialty to identify surgical re-
Discussion finements that could potentially reduce complication rates
and improve outcomes.
Describing hypospadias based on the position of the ure- The concept that factors other than meatal position may
thral opening has been suggested as the most reliable and impact surgical outcomes is not new and others have made
reproducible way to classify the anomaly [1]. Indeed, in efforts to investigate their surgical relevance. Sarhan and
most outcomes studies, patients are grouped according to colleagues evaluated the effect of specific urethral plate
meatal position (i.e. distal, proximal) [2e5]. However, characteristics on surgical outcomes in a study involving 80
given that hypospadias exists as a complex, other factors boys [7]. Although they did not standardize their observa-
may have as much impact on surgical outcomes as the po- tions according to surgical technique, of the factors eval-
sition of the urethral meatus. To our knowledge there is no uated, a urethral plate width of less than 8 mm was the
universally accepted method for classifying the severity of only one that correlated to the risk of a surgical compli-
the hypospadias complex in a standardized fashion. If one cation. Along the same lines, Holland et al. found a shallow
assumes that the severity of chordee, size of the glans, and urethral groove depth to be associated with meatal stenosis
the quality of the urethral plate impact surgical outcome, while a narrow urethral plate was associated with fistula
then it becomes difficult to compare one study to another formation [8]. In contrast, Nguyen et al. found no correla-
using only meatal position for stratifying patients. tion between urethral plate characteristics and risk of post-
Castagnetti and El-Ghoneimi emphasized the need for surgical complications [9]. Ziada et al. prospectively stud-
shared criteria for patient stratification in their 20-year ied the surgical outcomes of 3 different age groups under-
review of the management of severe hypospadias [2]. They going surgery for mid-shaft and distal hypospadias [10]. In
point out that a clear definition of severe hypospadias is addition to age and meatal position they also collected data
regarding urethral plate width, urethral wall thickness, and
the severity of chordee. Analysis to determine how these
factors impacted surgical outcomes was not included in
their study, but it highlights the sentiment that features of
the hypospadias complex other than meatal position are of
importance.
In response to the need for a uniformly accepted clas-
sification system for hypospadias, we developed the GMS
hypospadias score. Our goal was to develop a standardized
method for scoring the severity of the hypospadias com-
plex, which includes, but is not limited to, meatal position.
The criteria chosen were based on the anatomic features of
hypospadias felt most likely to impact complication rates as
well as the cosmetic and functional outcomes of surgical
repair. This classification scheme was developed with the
intent that it would be easy to use, reproducible, as
Figure 4 GMS scores of the study cohort. Note that patients objective as possible, and directly reflect the risk of a
who had a complication are represented by the red portion of surgical complication. The results of our study suggest that
the bar. (For interpretation of the references to color in this the GMS scoring method has high inter-observer reliability
figure legend, the reader is referred to the web version of this and also appears to correlate with the risk of a surgical
article.) complication. The average GMS score was significantly

Please cite this article in press as: Merriman LS, et al., The GMS hypospadias score: Assessment of inter-observer reliability and corre-
lation with post-operative complications, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/j.jpurol.2013.04.006
+ MODEL
The GMS hypospadias score 5

higher for those who developed a complication when most instances this was apparent on exam and taken into
compared to those who did not. Furthermore, an increased consideration by the rating surgeons. We also make a no-
risk of a surgical complication was noted when the total tation for the mega-meatus intact prepuce (MIP) variant of
GMS score was greater than 6. Although a more detailed hypospadias since repair in these patients may pose unique
and comprehensive grading system might be more prog- challenges.
nostic, it would have the potential disadvantage of being Finally, our study was conducted at a single institution
overly cumbersome and not practical for use in an office by a relatively small group of surgeons who were familiar
setting. with the grading scale. However, we noted a very high
One criticism of the GMS method is that the G score re- degree of inter-observer agreement even initially and with
mains subjective and actually evaluates two features of the new observer pairs, suggesting broader application of the
hypospadias complex, the glans and urethral plate. In scale is feasible. While care was taken to avoid observer
retrospect it may more be more practical to consider the bias, the possibility of this exists and external validation of
glans and urethral plate separately (i.e. GUMS). Currently, the GMS scale will therefore be necessary. Conflicting
data regarding glans size and a U component to the scale are operating room schedules also made it impractical for every
being collected to determine if and how this additional in- patient to be evaluated by two attending urologists, which
formation relates to surgical outcomes. Despite the sub- would otherwise be ideal.
jective nature of the G score, we feel that features of the
glans and urethral plate which can be observed but not easily
measured, are of critical importance. Such features include
Conclusions
the apparent thickness, elasticity, and quality of the
epithelium of the urethral plate. One could also foresee A standardized method for describing the severity of the
scenarios where measurements might actually be hypospadias complex would be useful for our discipline.
misleading. For example, a 6-month-old and 3-year-old with Although refinements or additions may be necessary, the
the same urethral plate width may have different risks for a GMS score provides a concise and reproducible way to
complication. The same issues could be encountered for describe the severity of hypospadias and correlates to the
patients of different weights or who have different penile risk of a surgical complication. This or a similar method may
sizes. Furthermore, assessment becomes increasingly com- prove useful for academic discussion, hypospadias
plex as measurements are obtained. For example, should the research, and parental counseling. Ongoing study with a
urethral plate be measured where it is widest, narrowest, or larger number of patients, a longer follow-up period, and
should an average be used? How is the concavity or convexity more detailed statistical analysis may enable the identifi-
of the urethral plate taken into consideration? Similarly, cation specific characteristics that correlate to the risk of a
should the glans be measured at its base, at the mid-glans, or surgical complication.
should a ratio be used? Again, the GMS classification system is
designed to be easy to use even in the office setting and it Conflict of interest
may prove to be as informative as a similar scale that in-
cludes objective measurements. Other grading systems None.
including The International Reflux Study Committee system
for grading urinary reflux and the Bosniak classification sys-
tem for grading renal cysts are also partially subjective and Funding
do not include measurements, yet have proven very useful
clinically. Indeed, we were impressed that a group of 7 pe- None.
diatric urologists with different training and backgrounds,
and blinded to one another’s assessment, had very high
agreement on what was a favorable, adequate, marginal, References
and unfavorable G score. This is a case where a trained eye
may be able to better assess, or at least categorize, features [1] Snodgrass W, Macedo A, Hoebeke P, Mouriquand PD. Hypo-
spadias dilemmas: a round table. J Pediatr Urol 2011;7:
and proportions that are difficult to concisely measure.
145e57.
Certainly refinements maybe necessary over time and with [2] Castagnetti M, El-Ghoneimi A. Surgical management of pri-
further investigation, but the current GMS classification mary severe hypospadias in children: systematic 20-year re-
system provides a starting point for studies related to view. J Urol 2010;184:1469e74.
developing a standardized instrument for scoring the [3] Rynja SP, Wouters GA, Van Schaijk M, Kok ET, De Jong TP, De
severity of hypospadias. Indeed, we have become accus- Kort LM. Long-term followup of hypospadias: functional and
tomed to using a patient’s G, M, and S scores as a rapid cosmetic results. J Urol 2009;182:1736e43.
reference point for assessing cosmetic and functional out- [4] Snodgrass W, Bush N, Cost N. Tubularized incised plate
comes following surgery. hypospadias repair for distal hypospadias. J Pediatr Urol 2010;
Limitations of the GMS method include the fact that 6(4):408e13.
[5] Frimberger D, Campbell J, Kropp BP. Hypospadias outcome in
associated scrotal anomalies, such as peno-scrotal trans-
the first 3 years after completing a pediatric urology fellow-
position, are not evaluated. Whether and how this should ship. J Pediatr Urol 2008;4(4):270e4.
be included in a standardized scoring method is a subject of [6] Giannantoni A. Hypospadias classification and repair: the riddle
debate. We have also encountered patients with severe of the sphinx. Eur Urol 2011;60:1190e1 [discussion 1e2].
urethral hypoplasia such that the surgical meatus is found [7] Sarhan O, Saad M, Helmy T, Hafez A. Effect of suturing
to be significantly proximal to the anatomic meatus. In technique and urethral plate characteristics on

Please cite this article in press as: Merriman LS, et al., The GMS hypospadias score: Assessment of inter-observer reliability and corre-
lation with post-operative complications, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/j.jpurol.2013.04.006
+ MODEL
6 L.S. Merriman et al.

complication rate following hypospadias repair: a pro- [9] Nguyen MT, Snodgrass WT, Zaontz MR. Effect of urethral plate
spective randomized study. J Urol 2009;182:682e5 [dis- characteristics on tubularized incised plate urethroplasty. J
cussion 5e6]. Urol 2004;171:1260e2 [discussion 2].
[8] Holland AJ, Smith GH. Effect of the depth and width of the [10] Ziada A, Hamza A, Abdel-Rassoul M, Habib E, Mohamed A,
urethral plate on tubularized incised plate urethroplasty. J Daw M. Outcomes of hypospadias repair in older children: a
Urol 2000;164:489e91. prospective study. J Urol 2011;185:2483e5.

Please cite this article in press as: Merriman LS, et al., The GMS hypospadias score: Assessment of inter-observer reliability and corre-
lation with post-operative complications, Journal of Pediatric Urology (2013), http://dx.doi.org/10.1016/j.jpurol.2013.04.006

You might also like