- Email: [email protected]
Formulation and validation of meatal stenosis grading system
Journal Pre-proof Formulation and validation of Meatal Stenosis Grading System Matan Mekayten, Eyal Meir, Ben-Chaim Jacob, Ezekiel H. Landau, Antoine E. Khoury, Ofer N. Gofrit, Mordechai duvdevani, Guy Hidas PII:
S1477-5131(19)30412-7
DOI:
https://doi.org/10.1016/j.jpurol.2019.11.012
Reference:
JPUROL 3328
To appear in:
Journal of Pediatric Urology
Received Date: 27 July 2019 Accepted Date: 23 November 2019
Please cite this article as: Mekayten M, Meir E, Jacob B-C, Landau EH, Khoury AE, Gofrit ON, duvdevani M, Hidas G, Formulation and validation of Meatal Stenosis Grading System, Journal of Pediatric Urology, https://doi.org/10.1016/j.jpurol.2019.11.012. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Published by Elsevier Ltd on behalf of Journal of Pediatric Urology Company.
Formulation and validation of Meatal Stenosis Grading System Matan Mekaytena, Eyal Meir a, Ben-Chaim Jacobb, Ezekiel H. Landau a, Antoine E. Khouryc, Ofer N. Gofrita, Mordechai duvdevani a and Guy Hidasa a
Hadassah Hebrew University Medical Center, Jerusalem, Israel, bTel Aviv Sourasky Medical Center, Tel Aviv, Israel, cCHOC Children's Hospital of Orange County, Orange, CA, USA,
Corresponding Author Matan Mekayten Pediatric Urology Unit Hadassah Hebrew University Medical Center, Jerusalem, Israel 91120 Mobile +972-52-6-807-807 Email – [email protected] Tel - 9722 6776874 Fax - 9722 6430929
Eyal Meir Pediatric Urology Unit Hadassah Hebrew University Medical Center, [email protected]
Ben-Chaim Jacob Tel Aviv Sourasky Medical Center, Paediatric UroIogy Unit, Department of Urology, Tel Aviv, Israel [email protected]
Ezekiel H. Landau Pediatric Urology Unit Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Antoine E. Khoury
[email protected] Department of Urology, University of California, Irvine, CA, USA CHOC Children's Hospital of Orange County, Orange, CA, USA
Ofer N. Gofrit Department of Urology Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Mordechai duvdevani Department of Urology Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Guy Hidas Pediatric Urology Unit Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Formulation and validation of Meatal Stenosis Grading System summary Introduction Meatal stenosis (MS) is a common finding in circumcised children. Indication for surgical correction are based on urinary symptoms as strength and direction of urine stream as well as physical examination, including direction and caliber of the urinary stream. There is no objective grading of MS severity and therefore indication for surgery and management protocols are vague. Objective We aimed to formulate a standardized, validated and reliable grading system for MS severity based on physical examination finding. Study Design Photographs of the urethral meatus were taken in patients scheduled for meatotomy due to MS whereas patients without this condition served as control. The photographs were rated by three experienced fellowship trained pediatric urologists. The study was conducted in two phases: 1) Development of a grading system by the expert panel. 2) Testing of the proposed grading system for inter- and intra-rater reliability. To estimate the correlation between different rates the Intra-Class Correlation Coefficient (ICC) was calculated. Results Three grades were generated: Grade 0 (wide open meatus, visible mucosa) ,Grade 1 (minimal mucosa/fibrotic tissue visible), and Grade 2 (pinpoint meatus/no mucosa visible/ large fibrotic layer). A panel of 51 raters (Pediatric urologist, community urologist, pediatricians) participated in the survey evaluating representative photos from
1
86 patients. Inter-rater reliability was high ICC=0.99[95% CI of 0.983-0.996, p<0.0001] Cronbach's Alpha =0.992. Eighteen raters participated in the same survey two weeks later for intra-rater reliability. An identical grading was obtained in 83.3% of photographs [Kappa=0.455(p<0.05)]. Conclusion We propose a grading system that is a valid, reliable, and reproducible method to classify the severity of MS on physical exam. This grading system could improve the healthcare provider's and parent's communication and can be a building block for further research in this field. A further research should assess the correlation with clinical signs and symptoms.
2
The Generated Meatal Stenosis Grading System Grade 0 – open meatus + mucosa seen + no web seen
Grade 1 – open meatus + mild mucosa seen + web seen
Grade 2 – pinpoint meatus + no mucosa seen + large web seen
3
Keywords meatal stenosis, pediatric urology, stricture, stream deviation, grading system
Introduction Meatal stenosis is clinically defined as narrowing of the urethral orifice[1], [2]. This condition occurs almost exclusively in circumcised infants, with a relatively high incidence reported as high as 5-20%[3]. Considerable portion of patients are asymptomatic and diagnosed by a pediatrician or a pediatric urologist on physical exam. Symptomatic patients usually present with upward deviation of urine stream, highvelocity narrow stream, prolonged voiding, penile pain during initiation of micturition, difficulty-to-aim, and rarely hematuria, urinary tract infections, straining while urinating, dribbling, urinary urgency and frequency[4], [5]. Physical examination is varied. Previous studies report a normal meatus size of 3.69 to 5.4 mm in average measured dorso-ventraly[6-7]. On physical examination, stenosis of meatus could range from mild narrowing to pin point opening with obstructing ventral webbing tissue[4]. If a child is watched while urinating an upward, thin and forceful stream will be observed with possibly incomplete bladder emptying[1]. The literature supports meatotomy or meatoplasty in meatal stenosis since if left untreated, this condition may lead to recurrent urinary tract infections and occasionally bladder complications[2], [8] But is it necessary to operate on all patients? Or only on selected cases? If yes, what can be conceded severe meatal stenosis? Will it be defined by the history? by physical examination? or by other diagnostic tests like uroflowmetry and post void residual? Grading systems are a common method used in different fields of medicine, including pediatric urology. Well-known examples of this are the prenatal 4
hydronephrosis grading system of the Society of Fetal Urology, the vesicoureteral reflux grades[9], [10] and the varicocele grading classification[11]. Grading systems facilitate common terminology and communication between pediatricians, pediatric urologists and parents. A grading system will objectively classify severity for proper prognosis, and to monitor treatment modality effect. Grading systems will also allow us to improve our methodology of future research in the field. In this study we aimed to develop a standardized, validated and reliable grading system for meatal stenosis severity based on physical examination. We anticipate that this standardized grading system will be a building block for future communication and research.
Material and Methods Patients and photographic imaging: Patients scheduled for meatotomy due to meatal stenosis were prospectively enrolled into the study. We included all patients aged above 3 years old (mean =6.76 SD 3.49) and excluded patients with other urinary anomalies or developmental delay. A control group was generated from the same age patients who came to our office with a chief complaint other than voiding dysfunction. Since meatal stenosis is rare in the uncircumcised patients' population, all patients were circumcised and toilet trained. Study protocol was approved by the local institutional review committee and parents signed an informed consent before recruited to the study
5
After signing an informed consent by the patient or his parents, two photographs of the urethral meatus, while closed and wide open, were taken. Psychometric methodology: The study consisted of two steps (Figure 1): 1. The methodological development of a grading system for MS. This includes developing the appropriate terms to describe MS and defining the descriptions for each grade, 2. Validating the proposed grading system by confirming inter- and intra-rater reliability. 1st step Generate a domain of observables and definition of each grade Because there is not an established standard nomenclature for describing MS, the content for the grading system (which discriminates each severity level) had to be generated. Pictures of 30 patients represent all variable presentations of a MS (including the control group), were reviewed independently by an expert panel of 3 experience fellowship trained pediatric urologist (GH, JBH, EHL), and a domain of observable was generated. In order to document which parameters were correlated better to MS severity, each rater independently assigned a subjective grade of 0 (no MS), grade 1 (mild to moderate MS), grade 2 (Severe MS), to each photograph and was asked to use the terms 6
previously generated to describe the reason for their specific grade assignment. The responses were analyzed and the most common nomenclature for each grade were included in the definitions, which were formatted onto a definition sheet with a correlating photograph image. 2nd Step:
Inter and Intra-rater reliability In order to validate the MS grading criteria, an electronic survey was first administered to 51 raters that were not involved in the development of the definition sheet. The raters group included pediatric urologists, pediatric urology fellows and nurse practitioners as well as adult urologists. Each rater was given a copy of the proposed grading system with a sample picture of the meatus of each grade. The raters were asked to assign a grade for each of 20 pictures of meatus in the survey (inter-rater reliability). two weeks after taking the survey, the same raters were asked to take the same survey (intra-rater reliability).
Statistical analysis To estimate the correlation between different rates the Intra-Class Correlation Coefficient (ICC) was calculated. To estimate the intra-rater reliability, the Kappa coefficient was calculated.
Results Generate a domain of observables and definition of each grade
7
The most common nomenclature used by the expert panel to describe severity of MS was divided into two categories namely the description of the urethral mucosa and the fibrotic tissue layer covering the ventral meatus (meatal web) (Figure 2). The panel agreed upon three grades for MS severity (0=normal, 1, 2) since using four grades of severity caused ambiguity between the median grades. Grade 0 (visible mucosa, no fibrotic tissue layer covering of ventral meatus), Grade 1 (minimal mucosa visible, a fibrotic tissue visible), and Grade 2 (pinpoint meatus, no mucosa visible large fibrotic layer). (Figure 2)
Stage 2: Inter and intra-rater reliability Twenty best representative photographs were selected from 86 children who participated in the study (80 patients from the study group and 6 control group). 51 raters agreed to participate in the survey. Inter-rater reliability among the 51 raters was high, Intraclass Correlation Coefficient of 0.991, [95% CI of 0.983-0.996, p<0.0001] Cronbach's Alpha =0.992. Eighteen participant raters agreed to retake the same survey two weeks later for late Intra-rater reliability study. An identical grading was obtaining to each photograph in 83.3% of the time on average, and an average Kappa score was 0.455 with Chi-Square McNemar Test (p<0.05).
8
Discussion Meatal stenosis (International Classification of Diseases 10th edition, 2020, code N35.911[12]) is a common condition seen in the pediatric circumcised patient and has been traditionally described by pediatricians and urologist using the subjective terms “mild,” “moderate,” or “severe.” In this study, we formulated an objective grading system for MS severity using reliable methodology. Although the severity of MS is considered by most physicians to be a major factor in decision making for surgical treatment because of potential clinical implications, very few studies demonstrate this relation. This discrepancy between MS and symptoms might be attributed to non-standardized and great variation in meatal stenosis definition[13] most studies do not define meatal stenosis, while other uses meatal caliber, inspection of urinary stream caliber, symptoms, or other features[3], [4], [8]. Standardizing meatal stenosis definition is an important step toward promoting standardized data collection and providing appropriate treatment for patients. This study is limited by the methodological validation of using photographs to rate the severity grading instead of actual physical exam, this might be countered by the high number of raters participated and multiple data points
This paper focused on the psychometric methodology to generate a reliable MS grading system for the pediatric circumcised male population. Our objectives were to have an easy and a simple to use instrument to assess MS severity in clinical and research scenarios. Using this grading system, we can define severity of MS objectively and efficiently. This is important for two significant reasons: 1) clinicians and parents
9
can reliably communicate about the severity of MS and no longer rely on subjective personal criteria to describe the level of stenosis, and 2) it is now possible to perform different studies using MS grade as a validated parameter. This grading system which is based on meatal appearance, clinical correlation (as abnormal urine stream and relevant complaints) cannot be skipped, and the two should be used in combination.
Conclusion The proposed grading system provides a valid, reliable, and reproducible method to classify the severity of meatal stenosis on physical exam. This grading system could improve healthcare providers and parents' communication. The correlation between grading severity clinical symptoms and indication for surgery is yet to be examined.
Conflict of interest None.
Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
References [1]
M.-H. Wang, “Surgical Management of Meatal Stenosis with Meatoplasty,” J. Vis. Exp. JoVE, no. 45, Nov. 2010.
[2]
M. F. Campbell, “Stenosis of the External Urethral Meatus11Presented at annual meeting, American Association of Genito-Urinary Surgeons, Stockbridge Mass., June 11, 1943.,” J. Urol., Dec. 1943.
10
[3]
M. Frisch and J. Simonsen, “Cultural background, non-therapeutic circumcision and the risk of meatal stenosis and other urethral stricture disease: Two nationwide registerbased cohort studies in Denmark 1977-2013,” Surg. J. R. Coll. Surg. Edinb. Irel., vol. 16, no. 2, pp. 107–118, Apr. 2018.
[4]
B. J. Morris and J. N. Krieger, “Re: Cultural background, non-therapeutic circumcision and the risk of meatal stenosis and other urethral stricture disease: Two nationwide register-based cohort studies in Denmark 1977–2013,” The Surgeon, vol. 16, no. 2, pp. 126–129, Apr. 2018.
[5]
“Clinical presentation and pathophysiology of meatal stenosis following circumcision Persad - 1995 - British Journal of Urology - Wiley Online Library.” [Online]. Available: https://onlinelibrary.wiley.com/doi/abs/10.1111/j.1464-410X.1995.tb07242.x. [Accessed: 02-Apr-2019].
[6]
G. S. Bhat, M. Shivalingiah, G. G. Nelivigi, and C. Ratkal, “The size of external urethral meatus on maximum stretch in Indian adult males.,” Indian J. Surg., vol. 76, no. 1, pp. 85–89, Feb. 2014.
[7]
M. Orkiszewski, J. Madej, and T. Kilian, “How wide a urethra should we produce?,” J. Pediatr. Urol., vol. 2, no. 5, pp. 473–476, Oct. 2006.
[8]
B. J. Morris and J. N. Krieger, “Does Circumcision Increase Meatal Stenosis Risk?—A Systematic Review and Meta-analysis,” Urology, vol. 110, pp. 16–26, Dec. 2017.
[9]
S. K. Fernbach, M. Maizels, and J. J. Conway, “Ultrasound grading of hydronephrosis: Introduction to the system used by the society for fetal urology,” Pediatr. Radiol., vol. 23, no. 6, pp. 478–480, Oct. 1993.
[10]
R. L. Lebowitz, H. Olbing, K. V. Parkkulainen, J. M. Smellie, and T. E. Tamminen-Möbius, “International system of radiographic grading of vesicoureteric reflux,” Pediatr. Radiol., vol. 15, no. 2, pp. 105–109, Feb. 1985.
11
[11]
L. Dubin and R. D. Amelar, “Varicocele Size And Results of Varicocelectomy in Selected Subfertile Men with Varicocele*,” Fertil. Steril., vol. 21, no. 8, pp. 606–609, Aug. 1970.
[12]
“2020 ICD-10-CM Diagnosis Code N35.911: Unspecified urethral stricture, male, meatal.” [Online]. Available: https://www.icd10data.com/ICD10CM/Codes/N00N99/N30-N39/N35-/N35.911. [Accessed: 11-Oct-2019].
[13]
B. J. Morris, S. Moreton, and J. N. Krieger, “Meatal stenosis: getting the diagnosis right,” Res. Rep. Urol., vol. 10, pp. 237–239, Dec. 2018.
12
Figure 1: Psychometric Methodology Process to Generate Meatal Stenosis Grading System
Step 1: Development of grading system • Generate a domain of observables • Definition of each grade
Step 2: Validation of grading system • Inter-rater reliablity • Intra-rater reliablity
13
Figure 2 - The Generated Meatal Stenosis Grading System Grade 0 – open meatus + mucosa seen + no web seen
Grade 1 – open meatus + mild mucosa seen + web seen
Grade 2 – pinpoint meatus + no mucosa seen + large web seen
14
Legends of figures: Figure 1: Psychometric Methodology Process to Generate Meatal Stenosis Grading System. Figure 2: The Generated Meatal Stenosis Grading System.
S1477-5131(19)30412-7
DOI:
https://doi.org/10.1016/j.jpurol.2019.11.012
Reference:
JPUROL 3328
To appear in:
Journal of Pediatric Urology
Received Date: 27 July 2019 Accepted Date: 23 November 2019
Please cite this article as: Mekayten M, Meir E, Jacob B-C, Landau EH, Khoury AE, Gofrit ON, duvdevani M, Hidas G, Formulation and validation of Meatal Stenosis Grading System, Journal of Pediatric Urology, https://doi.org/10.1016/j.jpurol.2019.11.012. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Published by Elsevier Ltd on behalf of Journal of Pediatric Urology Company.
Formulation and validation of Meatal Stenosis Grading System Matan Mekaytena, Eyal Meir a, Ben-Chaim Jacobb, Ezekiel H. Landau a, Antoine E. Khouryc, Ofer N. Gofrita, Mordechai duvdevani a and Guy Hidasa a
Hadassah Hebrew University Medical Center, Jerusalem, Israel, bTel Aviv Sourasky Medical Center, Tel Aviv, Israel, cCHOC Children's Hospital of Orange County, Orange, CA, USA,
Corresponding Author Matan Mekayten Pediatric Urology Unit Hadassah Hebrew University Medical Center, Jerusalem, Israel 91120 Mobile +972-52-6-807-807 Email – [email protected] Tel - 9722 6776874 Fax - 9722 6430929
Eyal Meir Pediatric Urology Unit Hadassah Hebrew University Medical Center, [email protected]
Ben-Chaim Jacob Tel Aviv Sourasky Medical Center, Paediatric UroIogy Unit, Department of Urology, Tel Aviv, Israel [email protected]
Ezekiel H. Landau Pediatric Urology Unit Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Antoine E. Khoury
[email protected] Department of Urology, University of California, Irvine, CA, USA CHOC Children's Hospital of Orange County, Orange, CA, USA
Ofer N. Gofrit Department of Urology Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Mordechai duvdevani Department of Urology Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Guy Hidas Pediatric Urology Unit Hadassah Hebrew University Medical Center, Jerusalem, Israel [email protected]
Formulation and validation of Meatal Stenosis Grading System summary Introduction Meatal stenosis (MS) is a common finding in circumcised children. Indication for surgical correction are based on urinary symptoms as strength and direction of urine stream as well as physical examination, including direction and caliber of the urinary stream. There is no objective grading of MS severity and therefore indication for surgery and management protocols are vague. Objective We aimed to formulate a standardized, validated and reliable grading system for MS severity based on physical examination finding. Study Design Photographs of the urethral meatus were taken in patients scheduled for meatotomy due to MS whereas patients without this condition served as control. The photographs were rated by three experienced fellowship trained pediatric urologists. The study was conducted in two phases: 1) Development of a grading system by the expert panel. 2) Testing of the proposed grading system for inter- and intra-rater reliability. To estimate the correlation between different rates the Intra-Class Correlation Coefficient (ICC) was calculated. Results Three grades were generated: Grade 0 (wide open meatus, visible mucosa) ,Grade 1 (minimal mucosa/fibrotic tissue visible), and Grade 2 (pinpoint meatus/no mucosa visible/ large fibrotic layer). A panel of 51 raters (Pediatric urologist, community urologist, pediatricians) participated in the survey evaluating representative photos from
1
86 patients. Inter-rater reliability was high ICC=0.99[95% CI of 0.983-0.996, p<0.0001] Cronbach's Alpha =0.992. Eighteen raters participated in the same survey two weeks later for intra-rater reliability. An identical grading was obtained in 83.3% of photographs [Kappa=0.455(p<0.05)]. Conclusion We propose a grading system that is a valid, reliable, and reproducible method to classify the severity of MS on physical exam. This grading system could improve the healthcare provider's and parent's communication and can be a building block for further research in this field. A further research should assess the correlation with clinical signs and symptoms.
2
The Generated Meatal Stenosis Grading System Grade 0 – open meatus + mucosa seen + no web seen
Grade 1 – open meatus + mild mucosa seen + web seen
Grade 2 – pinpoint meatus + no mucosa seen + large web seen
3
Keywords meatal stenosis, pediatric urology, stricture, stream deviation, grading system
Introduction Meatal stenosis is clinically defined as narrowing of the urethral orifice[1], [2]. This condition occurs almost exclusively in circumcised infants, with a relatively high incidence reported as high as 5-20%[3]. Considerable portion of patients are asymptomatic and diagnosed by a pediatrician or a pediatric urologist on physical exam. Symptomatic patients usually present with upward deviation of urine stream, highvelocity narrow stream, prolonged voiding, penile pain during initiation of micturition, difficulty-to-aim, and rarely hematuria, urinary tract infections, straining while urinating, dribbling, urinary urgency and frequency[4], [5]. Physical examination is varied. Previous studies report a normal meatus size of 3.69 to 5.4 mm in average measured dorso-ventraly[6-7]. On physical examination, stenosis of meatus could range from mild narrowing to pin point opening with obstructing ventral webbing tissue[4]. If a child is watched while urinating an upward, thin and forceful stream will be observed with possibly incomplete bladder emptying[1]. The literature supports meatotomy or meatoplasty in meatal stenosis since if left untreated, this condition may lead to recurrent urinary tract infections and occasionally bladder complications[2], [8] But is it necessary to operate on all patients? Or only on selected cases? If yes, what can be conceded severe meatal stenosis? Will it be defined by the history? by physical examination? or by other diagnostic tests like uroflowmetry and post void residual? Grading systems are a common method used in different fields of medicine, including pediatric urology. Well-known examples of this are the prenatal 4
hydronephrosis grading system of the Society of Fetal Urology, the vesicoureteral reflux grades[9], [10] and the varicocele grading classification[11]. Grading systems facilitate common terminology and communication between pediatricians, pediatric urologists and parents. A grading system will objectively classify severity for proper prognosis, and to monitor treatment modality effect. Grading systems will also allow us to improve our methodology of future research in the field. In this study we aimed to develop a standardized, validated and reliable grading system for meatal stenosis severity based on physical examination. We anticipate that this standardized grading system will be a building block for future communication and research.
Material and Methods Patients and photographic imaging: Patients scheduled for meatotomy due to meatal stenosis were prospectively enrolled into the study. We included all patients aged above 3 years old (mean =6.76 SD 3.49) and excluded patients with other urinary anomalies or developmental delay. A control group was generated from the same age patients who came to our office with a chief complaint other than voiding dysfunction. Since meatal stenosis is rare in the uncircumcised patients' population, all patients were circumcised and toilet trained. Study protocol was approved by the local institutional review committee and parents signed an informed consent before recruited to the study
5
After signing an informed consent by the patient or his parents, two photographs of the urethral meatus, while closed and wide open, were taken. Psychometric methodology: The study consisted of two steps (Figure 1): 1. The methodological development of a grading system for MS. This includes developing the appropriate terms to describe MS and defining the descriptions for each grade, 2. Validating the proposed grading system by confirming inter- and intra-rater reliability. 1st step Generate a domain of observables and definition of each grade Because there is not an established standard nomenclature for describing MS, the content for the grading system (which discriminates each severity level) had to be generated. Pictures of 30 patients represent all variable presentations of a MS (including the control group), were reviewed independently by an expert panel of 3 experience fellowship trained pediatric urologist (GH, JBH, EHL), and a domain of observable was generated. In order to document which parameters were correlated better to MS severity, each rater independently assigned a subjective grade of 0 (no MS), grade 1 (mild to moderate MS), grade 2 (Severe MS), to each photograph and was asked to use the terms 6
previously generated to describe the reason for their specific grade assignment. The responses were analyzed and the most common nomenclature for each grade were included in the definitions, which were formatted onto a definition sheet with a correlating photograph image. 2nd Step:
Inter and Intra-rater reliability In order to validate the MS grading criteria, an electronic survey was first administered to 51 raters that were not involved in the development of the definition sheet. The raters group included pediatric urologists, pediatric urology fellows and nurse practitioners as well as adult urologists. Each rater was given a copy of the proposed grading system with a sample picture of the meatus of each grade. The raters were asked to assign a grade for each of 20 pictures of meatus in the survey (inter-rater reliability). two weeks after taking the survey, the same raters were asked to take the same survey (intra-rater reliability).
Statistical analysis To estimate the correlation between different rates the Intra-Class Correlation Coefficient (ICC) was calculated. To estimate the intra-rater reliability, the Kappa coefficient was calculated.
Results Generate a domain of observables and definition of each grade
7
The most common nomenclature used by the expert panel to describe severity of MS was divided into two categories namely the description of the urethral mucosa and the fibrotic tissue layer covering the ventral meatus (meatal web) (Figure 2). The panel agreed upon three grades for MS severity (0=normal, 1, 2) since using four grades of severity caused ambiguity between the median grades. Grade 0 (visible mucosa, no fibrotic tissue layer covering of ventral meatus), Grade 1 (minimal mucosa visible, a fibrotic tissue visible), and Grade 2 (pinpoint meatus, no mucosa visible large fibrotic layer). (Figure 2)
Stage 2: Inter and intra-rater reliability Twenty best representative photographs were selected from 86 children who participated in the study (80 patients from the study group and 6 control group). 51 raters agreed to participate in the survey. Inter-rater reliability among the 51 raters was high, Intraclass Correlation Coefficient of 0.991, [95% CI of 0.983-0.996, p<0.0001] Cronbach's Alpha =0.992. Eighteen participant raters agreed to retake the same survey two weeks later for late Intra-rater reliability study. An identical grading was obtaining to each photograph in 83.3% of the time on average, and an average Kappa score was 0.455 with Chi-Square McNemar Test (p<0.05).
8
Discussion Meatal stenosis (International Classification of Diseases 10th edition, 2020, code N35.911[12]) is a common condition seen in the pediatric circumcised patient and has been traditionally described by pediatricians and urologist using the subjective terms “mild,” “moderate,” or “severe.” In this study, we formulated an objective grading system for MS severity using reliable methodology. Although the severity of MS is considered by most physicians to be a major factor in decision making for surgical treatment because of potential clinical implications, very few studies demonstrate this relation. This discrepancy between MS and symptoms might be attributed to non-standardized and great variation in meatal stenosis definition[13] most studies do not define meatal stenosis, while other uses meatal caliber, inspection of urinary stream caliber, symptoms, or other features[3], [4], [8]. Standardizing meatal stenosis definition is an important step toward promoting standardized data collection and providing appropriate treatment for patients. This study is limited by the methodological validation of using photographs to rate the severity grading instead of actual physical exam, this might be countered by the high number of raters participated and multiple data points
This paper focused on the psychometric methodology to generate a reliable MS grading system for the pediatric circumcised male population. Our objectives were to have an easy and a simple to use instrument to assess MS severity in clinical and research scenarios. Using this grading system, we can define severity of MS objectively and efficiently. This is important for two significant reasons: 1) clinicians and parents
9
can reliably communicate about the severity of MS and no longer rely on subjective personal criteria to describe the level of stenosis, and 2) it is now possible to perform different studies using MS grade as a validated parameter. This grading system which is based on meatal appearance, clinical correlation (as abnormal urine stream and relevant complaints) cannot be skipped, and the two should be used in combination.
Conclusion The proposed grading system provides a valid, reliable, and reproducible method to classify the severity of meatal stenosis on physical exam. This grading system could improve healthcare providers and parents' communication. The correlation between grading severity clinical symptoms and indication for surgery is yet to be examined.
Conflict of interest None.
Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
References [1]
M.-H. Wang, “Surgical Management of Meatal Stenosis with Meatoplasty,” J. Vis. Exp. JoVE, no. 45, Nov. 2010.
[2]
M. F. Campbell, “Stenosis of the External Urethral Meatus11Presented at annual meeting, American Association of Genito-Urinary Surgeons, Stockbridge Mass., June 11, 1943.,” J. Urol., Dec. 1943.
10
[3]
M. Frisch and J. Simonsen, “Cultural background, non-therapeutic circumcision and the risk of meatal stenosis and other urethral stricture disease: Two nationwide registerbased cohort studies in Denmark 1977-2013,” Surg. J. R. Coll. Surg. Edinb. Irel., vol. 16, no. 2, pp. 107–118, Apr. 2018.
[4]
B. J. Morris and J. N. Krieger, “Re: Cultural background, non-therapeutic circumcision and the risk of meatal stenosis and other urethral stricture disease: Two nationwide register-based cohort studies in Denmark 1977–2013,” The Surgeon, vol. 16, no. 2, pp. 126–129, Apr. 2018.
[5]
“Clinical presentation and pathophysiology of meatal stenosis following circumcision Persad - 1995 - British Journal of Urology - Wiley Online Library.” [Online]. Available: https://onlinelibrary.wiley.com/doi/abs/10.1111/j.1464-410X.1995.tb07242.x. [Accessed: 02-Apr-2019].
[6]
G. S. Bhat, M. Shivalingiah, G. G. Nelivigi, and C. Ratkal, “The size of external urethral meatus on maximum stretch in Indian adult males.,” Indian J. Surg., vol. 76, no. 1, pp. 85–89, Feb. 2014.
[7]
M. Orkiszewski, J. Madej, and T. Kilian, “How wide a urethra should we produce?,” J. Pediatr. Urol., vol. 2, no. 5, pp. 473–476, Oct. 2006.
[8]
B. J. Morris and J. N. Krieger, “Does Circumcision Increase Meatal Stenosis Risk?—A Systematic Review and Meta-analysis,” Urology, vol. 110, pp. 16–26, Dec. 2017.
[9]
S. K. Fernbach, M. Maizels, and J. J. Conway, “Ultrasound grading of hydronephrosis: Introduction to the system used by the society for fetal urology,” Pediatr. Radiol., vol. 23, no. 6, pp. 478–480, Oct. 1993.
[10]
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Figure 1: Psychometric Methodology Process to Generate Meatal Stenosis Grading System
Step 1: Development of grading system • Generate a domain of observables • Definition of each grade
Step 2: Validation of grading system • Inter-rater reliablity • Intra-rater reliablity
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Figure 2 - The Generated Meatal Stenosis Grading System Grade 0 – open meatus + mucosa seen + no web seen
Grade 1 – open meatus + mild mucosa seen + web seen
Grade 2 – pinpoint meatus + no mucosa seen + large web seen
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Legends of figures: Figure 1: Psychometric Methodology Process to Generate Meatal Stenosis Grading System. Figure 2: The Generated Meatal Stenosis Grading System.