Abstract

Background

The introduction of bleeding assessment tools (BATs) to quantify the presence and severity of commonly reported bleeding symptoms has received increased interest over the past decade. Bleeding scores, along with laboratory data and family history, can assist the clinician in the assessment of a suspected mild bleeding disorder (MBD). While clinician-administered BATs have been utilized frequently, implementation and validation of the accuracy of a self-report or parent-proxy BAT have yet to be investigated. The primary objective of this study was to determine the accuracy of a parent-administered BAT by measuring the level of agreement between parent and clinician responses to the Condensed MCMDM-1VWD Bleeding Questionnaire.

Methods

Our study population included children aged 0-19 years presenting to the hematology clinic at Nationwide Children's Hospital (Columbus, OH) for initial evaluation of a suspected MBD or for follow-up evaluation of a previously diagnosed MBD. At the time of the visit, the parent/caregiver completed a short demographic survey and a modified version (targeted for a 6th grade comprehension level) of the Condensed MCMDM-1VWD Bleeding Questionnaire. The treating provider also completed the BAT by interviewing the patient and his/her caregiver; clinicians were blinded to the results of the parent BAT. Both the parent and clinician versions of the BAT were scored and analyzed in the same manner for ease of comparison. We calculated the percentage of agreement and weighted kappa statistic for individual bleeding symptoms as well as the mean across all questionnaire items. We also examined the agreement between caregiver and clinician responses in regards to patient age, gender, diagnosis (new versus follow-up patient), and parent education level.

Results

To date, we have enrolled 55 eligible patients. The overall mean bleeding score (BS) as calculated from the parent-report BAT was 5.98 (range: -1-25), while the mean BS for the clinician-report BAT was 3.87 (range: 0-16). The mean percentage of agreement between parents and clinicians across all items was 76% (range: 58-98%). The mean weighted kappa statistic was 0.31 (range: -0.04-0.79), representing fair agreement (based on Landis and Koch criteria); the mean Gwet's AC1 (an alternative kappa statistic) was 0.72 (range: 0.48-0.98), representing substantial agreement. Overall, 20% of parent and clinician total bleeding scores matched exactly, and an additional 42% of parent and clinician scores varied by only one to two points. 82% of the study population had an abnormal total bleeding score (defined as ≥2) when rated by parents and 78% had an abnormal total score when rated by clinicians (82% agreement, kappa = 0.43, Gwet's AC1 = 0.73). Tests for equal kappa coefficients did not show significant differences in agreement between parents and clinicians when compared by patient gender, age, diagnosis, or parent education level.

Discussion

To our knowledge, the results of a patient and/or parent-administered BAT score have not been studied to determine their accuracy and feasibility of use as a screening method for patients with a suspected MBD. While parents tended to over-report bleeding as compared to clinicians, overall, parent and clinician bleeding scores were similar in our study, and these results lend support for the potential use of a modified proxy-report BAT in a clinic setting. Additional research into the construct of the parent-administered BAT is needed to further improve the accuracy of parent-reported bleeding symptoms.

Table I

Parent and clinician responses to the Condensed MCMDM-1VWD Bleeding Questionnaire.

Bleeding Symptom (Possible Range)Parent MeanClinician MeanPercentage of AgreementKappaGwet's AC1
Epistaxis (0-4) 1.02 1.05 71 0.65 0.66 
Cutaneous (0-2) 0.55 0.62 63 0.36 0.48 
Minor wounds (0-4) 0.80 0.35 71 0.23 0.67 
Oral cavity (0-4) 0.71 0.29 67 0.23 0.63 
Gastrointestinal bleeding (0-3) 0.15 0.11 91 0.40 0.90 
Tooth extraction (-1- 4) 0.49 0.07 71 0.26 0.68 
Surgery (-1-4) 0.42 0.13 67 0.22 0.63 
Menorrhagia (0-4) 2.45 1.64 58 0.59 0.48 
Muscle hematomas (0-4) 0.11 0.07 89 -0.04 0.88 
Hemarthrosis (0-4) 0.20 0.00 89 0.00 0.89 
Central nervous system bleeding (0,3-4) 0.16 0.11 98 0.79 0.98 
Total Bleeding Score (-2-41) 5.98 3.87 76* 0.31* 0.72* 
Bleeding Symptom (Possible Range)Parent MeanClinician MeanPercentage of AgreementKappaGwet's AC1
Epistaxis (0-4) 1.02 1.05 71 0.65 0.66 
Cutaneous (0-2) 0.55 0.62 63 0.36 0.48 
Minor wounds (0-4) 0.80 0.35 71 0.23 0.67 
Oral cavity (0-4) 0.71 0.29 67 0.23 0.63 
Gastrointestinal bleeding (0-3) 0.15 0.11 91 0.40 0.90 
Tooth extraction (-1- 4) 0.49 0.07 71 0.26 0.68 
Surgery (-1-4) 0.42 0.13 67 0.22 0.63 
Menorrhagia (0-4) 2.45 1.64 58 0.59 0.48 
Muscle hematomas (0-4) 0.11 0.07 89 -0.04 0.88 
Hemarthrosis (0-4) 0.20 0.00 89 0.00 0.89 
Central nervous system bleeding (0,3-4) 0.16 0.11 98 0.79 0.98 
Total Bleeding Score (-2-41) 5.98 3.87 76* 0.31* 0.72* 
*

Represents mean across all questionnaire items

Disclosures:

Lang:OSUCOM Bennett Medical Student Research Scholarship: Research Funding; ASH HONORS Award: Research Funding.

Author notes

*

Asterisk with author names denotes non-ASH members.