CLINICAL ARTICLE J Neurosurg Pediatr 26:483–489, 2020

Anxiety, depression, fatigue, and burden in the pediatric hydrocephalus population

Kathrin Zimmerman, BA,1 Bobby May, BS,2 Katherine Barnes, BS,1 Anastasia Arynchyna, MPH,1 Elizabeth N. Alford, MD,1 Caroline Arata Wessinger, BS,1 Laura Dreer, PhD,3 Inmaculada Aban, PhD,4 James M. Johnston, MD,1 Curtis J. Rozzelle, MD,1 Jeffrey P. Blount, MD,1 and Brandon G. Rocque, MD, MS1

1Department of , Division of Pediatrics; Departments of 3Psychology and 4Statistics, University of Alabama at Birmingham, Alabama; and 2School of Medicine, University of Mississippi Medical Center, Jackson, Mississippi

OBJECTIVE Childhood hydrocephalus is a common chronic medical condition. However, little is known about the bur- den of headache and psychological comorbidities in children living with hydrocephalus. The purpose of this study was to determine the prevalence and severity of these conditions among the pediatric hydrocephalus population. METHODS During routine neurosurgery clinic visits from July 2017 to February 2018, the authors administered four surveys to children ages 7 years and older: Pediatric Disability Assessment (PedMIDAS), Patient-Reported Outcomes Measurement Information System (PROMIS) Anxiety, PROMIS Depression, and PROMIS Fatigue. The PedMIDAS is an assessment of headache disability in pediatric and adolescent patients. The PROMIS measures are pediatric self-reported instruments to assess social and emotional health. PROMIS measures utilize T-scores (mean 50, SD 10) to compare anxiety, depression, and fatigue in specific populations to those in the US general population. Clinical and demographic data were collected from the medical record (hydrocephalus etiology, , race, etc.) and tested for associations with survey measure scores. RESULTS Forty children completed the PedMIDAS. Ten percent of them were in the severe headache range, 5% were in the moderate range, and 5% were in the mild range. There was a statistically significant association between undergo- ing a cluster of shunt operations and headache burden (p = 0.003). Forty children completed all three PROMIS measures. The mean anxiety score was 45.8 (SD 11.7), and 2.5% of children scored in the severe anxiety range, 17.5% in the moderate range, and 20% in the mild range. The mean depression score was 42.7 (SD 10.0), with 2.5% of children scoring in the severe depression range, 5% in the moderate range, and 12.5% in the mild range. The mean fatigue score was 45.1 (SD 16.4), with 15% percent of children scoring in the severe fatigue range, 10% in the moderate range, and 7.5% in the mild range. There were no statistically significant associations between child anxiety, depression, or fatigue and clinical or demographic variables. CONCLUSIONS Children with hydrocephalus have an average burden of headache, anxiety, depression, and fatigue as compared to the general population overall. Having a cluster of shunt operations correlates with a higher headache burden, but no clinical or demographic variable is associated with anxiety, depression, or fatigue. https://thejns.org/doi/abs/10.3171/2020.4.PEDS19697 KEYWORDS hydrocephalus; pediatrics; anxiety; depression; fatigue; headache

ydrocephalus is the most common condition their lives.2 Anecdotally, pediatric neurosurgeons have treated by pediatric neurosurgeons and the most noted that children with hydrocephalus may suffer from common reason for in children.1 chronic more commonly than those without HThere are an estimated one million people in the US with hydrocephalus. While headache is the most common hydrocephalus, the majority of whom were diagnosed in symptom of shunt malfunction or inadequately treated childhood and have lived with hydrocephalus for most of hydrocephalus, it can also be present in the absence of a

ABBREVIATIONS ETV = endoscopic third ; PedMIDAS = Pediatric Migraine Disability Assessment; PROMIS = Patient-Reported Outcomes Measurement Information System. SUBMITTED November 23, 2019. ACCEPTED April 30, 2020. INCLUDE WHEN CITING Published online July 24, 2020; DOI: 10.3171/2020.4.PEDS19697.

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Unauthenticated | Downloaded 09/28/21 02:30 AM UTC Zimmerman et al. shunt-related problem (malfunction, overdrainage, infec- tus, child sex, and child race were collected from the tion, etc.). However, there are no studies that assess the Psychosocial Assessment Tool 2.0 (PAT), administered burden of headache in children with hydrocephalus. to caregivers on the day of the clinic visit. Clinical data Similarly, children with chronic medical conditions on the etiology of hydrocephalus, number of shunt revi- can suffer from associated psychological comorbidities. sions, whether the child’s hydrocephalus was treated with The psychological and physical stress of a chronic medi- ETV alone (no history of CSF shunt), gestational age at cal condition can cause long-lasting psychosocial impact , distance from residence to hospital, history of shunt on patients and caregivers. Previous studies have exam- infection, and number of external ventricular drains in a ined the relationship between disease and psychological lifetime were collected from the electronic medical record comorbidities, such as depression, anxiety, and fatigue.3–12 and the prospectively maintained Hydrocephalus Clinical For example, a link has been shown between anxiety and Research Network (HCRN) institutional database. headache in patients with chronic migraine.13 However, Recognizing that shunts can fail repeatedly, we defined evaluation of the psychological comorbidities of pediatric a new variable to capture those patients who had a history hydrocephalus has not been performed. of a cluster of shunt revisions. A “shunt cluster” was de- The purpose of this study was to determine the preva- fined as undergoing three or more shunt operations within lence and severity of headache, depression, anxiety, and a 2-month period. The purpose of the cluster variable was fatigue in a sample of children with hydrocephalus. Un- to capture those patients who experienced a period of fre- derstanding these factors may play a crucial role in im- quent shunt failure and multiple operations. Furthermore, proving the care provided to children with hydrocephalus this allowed us to differentiate this group from patients and their families, enhancing outcomes and quality of life. who may have had a large number of shunt revisions but never a cluster of many within a short time. The cluster Methods variable was defined prior to data collection and analysis. Study Setting and Population Prior surgical history in the electronic medical record was reviewed to identify whether a patient had an identifiable From July 2017 to February 2018, we performed a shunt cluster. cross-sectional analysis of headache, anxiety, depression, and fatigue in children and adolescents with hydrocepha- PedMIDAS lus. Patients were enrolled during routine neurosurgical clinic visits at a large children’s hospital. Annually, the The PedMIDAS is a pediatric version (validated for ages 4–18) of the adult MIDAS survey that was created center follows over 600 patients with hydrocephalus and 15,16 performs approximately 450 operations for treatment of by Lipton and Stewart for adults ages 20–50 years. the disorder. The pediatric survey has six questions that ask how many Patients ages 7–21 years with surgically treated hy- days out of the last 3 months a child has been incapable drocephalus, (CSF) shunting, or en- of performing adequately in the areas of school, sports, doscopic third ventriculostomy (ETV) were eligible for and home activities (such as homework or chores). Re- enrollment. Eligibility criteria included the ability to in- sponses to the survey are added up and scored according dependently complete a study questionnaire, as assessed to the PedMIDAS grading scale: score ≤ 10, little to no by the patient’s caregiver and the research team. Children headache-related disability; score 11–30, mild disability; who did not have the maturity or mental capacity to com- score 31–50, moderate disability; and score > 50, severe plete the surveys were excluded. For the purposes of our disability due to headaches. For the analysis of their rela- research question, it was important to include only “well” tionship to clinical variables, the PedMIDAS scores were children, ensuring that the headache, anxiety, depression, categorized into a binary outcome (little/none vs mild/ and fatigue symptoms reported were unrelated to potential moderate/severe). This cutoff was chosen because of the shunt malfunction. Therefore, children who had under- small number of observations with scores > 0. gone a surgical procedure for their hydrocephalus within 30 days of the clinic visit were excluded. PROMIS Survey The PROMIS Anxiety, Depression, and Fatigue mea- Data Collection sures are a subset of surveys created within the Health- Approval for this study was obtained from the Insti- Measures program through Northwestern University.17 tutional Review Board of the University of Alabama at The format of these surveys asks patients how often they Birmingham. Informed consent and assent were obtained have experienced certain feelings (e.g., “I felt nervous”) as appropriate based on patient age. Study data were col- and asks them to assign a numerical value, with 1 indicat- lected and managed using Research Electronic Data Cap- ing never and 5 indicating almost always. These instru- ture (REDCap) tools hosted at the University of Alabama ments are scored on a T-score metric, with 50 being the at Birmingham.14 Four surveys were administered to study mean score in the general population with an SD of 10. A participants: Pediatric Migraine Disability Assessment score above 50 indicates a higher propensity to suffer from (PedMIDAS), Patient-Reported Outcomes Measurement the symptom being measured (depression, anxiety, and/ Information System (PROMIS) Anxiety, PROMIS De- or fatigue). According to the HealthMeasures program, pression, and PROMIS Fatigue. scores up to 0.5 SD above the mean (a score between 50 Data on the social factors of caregiver age, caregiver and 55) indicate mild symptoms or impairment compared marital status, caregiver education, child insurance sta- to the general population. Scores 0.5 to 1.5 SD above the

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Unauthenticated | Downloaded 09/28/21 02:30 AM UTC Zimmerman et al. mean (between 55 and 65) indicate moderate symptoms or TABLE 1. Summary of demographic and clinical variables in impairment, and a score higher than 1.5 SD (scaled score pediatric patients with hydrocephalus of 65 or higher) indicates severe symptoms or impairment. Variable Median (min, max) or No. (%) Statistical Analysis Age in yrs (n = 39) 13.5 (7, 21) The PedMIDAS results and the three PROMIS mea- Gestational age at birth in wks (n = 32) 37.5 (22, 41) sures were each analyzed for associations with categorical No. of procedures (n = 37) 2 (1, 35) clinical and demographical variables using the Kruskal- Distance from hospital in miles (n = 36) 78.75 (6.2, 202.0) Wallis test. The Spearman rank correlation was used to Sex (n = 40) analyze associations between survey scores and continu- Male 20 (50) ous demographic and clinical variables. To investigate the Female 20 (50) association between the PedMIDAS binary outcome and Race (n = 40) the PROMIS scores, we again used the Kruskal-Wallis White 25 (63) test. Missing clinical and demographic data are explained Non-white 15 (38) by the following: participant did not complete all survey Caregiver age in yrs (n = 38) measures; participant was not enrolled in the HCRN reg- 21 & over 33 (87) istry, so not all variables were available; and information Below 21 5 (13) was not available in the electronic medical record. A p value < 0.05 was considered significant. No adjust- Caregiver education (n = 38) ment due to multiple testing was performed, as this study College/grad school 18 (47) is exploratory: the intention of the study was to identify Other 20 (53) possible associations that will help determine what follow- Caregiver marital status (n = 38) up studies need to be conducted. All analyses were done in Married/partnered 21 (55) SAS version 9.4 (SAS Institute). Other 17 (45) Child insurance status (n = 37) Private 17 (46) Results Public 20 (54) Sixty patients were eligible for inclusion in the study, ETV alone (n = 36) and 40 children consented to participate. Patients who Yes 2 (6) were eligible for inclusion but chose not to participate of- No/none 34 (94) ten did so because of time constraints, as survey comple- No. of clusters (n = 35) tion required an average of 30 minutes and frequently re- None 29 (83) quired staying additional time after the provider visit con- At least 1 6 (17) cluded. No subjects were excluded after data collection. Twenty patients (50%) were male and 25 (63%) were non- Shunt infection (n = 37) Hispanic white. Among the primary caregivers, 33 (87%) None 30 (81) were at least 21 years old, 18 (47%) had received at least a At least 1 7 (19) college degree, and 21 (55%) were either married or part- No. of EVDs (n = 36) nered. The majority of the children (54%) were covered None 24 (67) by public insurance. The most common underlying etiolo- At least 1 12 (33) gies of hydrocephalus were myelomeningocele (22%) and Etiology of HCP (n = 37) intraventricular hemorrhage of prematurity (22%). Two Aqueductal 1 (3) patients (6%) had hydrocephalus treated with ETV alone. IVH 8 (22) Seven (18%) had experienced at least one shunt infection, MMC 8 (22) and 6 (15%) had experienced a cluster of hydrocephalus Other* 20 (54) . A summary of demographic and clinical vari- grad = graduate; HCP = hydrocephalus; IVH = intraventricular hemorrhage; ables is provided in Table 1. MMC = myelomeningocele; n = number of cases. Headache severity as measured by the PedMIDAS was * Postinfectious, 2 cases; spontaneous hemorrhage, 1 case; tumor found to have a mean score of 20.2 with an SD of 66.1 or other midbrain lesion, 3 cases; post–, 2 cases; posterior fossa (range 0–386.0). Thirty-two participants (80%) fell into cyst, 2 cases; communicating congenital hydrocephalus, 8 cases; unknown, 2 the little to no headache-related disability range. Ten per- cases. cent of children scored in the severe headache range, 5% scored in the moderate range, and 5% scored in the mild range. The three PROMIS measures for depression, anxi- ety, and fatigue were each scored separately. The PROMIS anxiety range, 17.5% in the moderate range, and 20% in Depression survey had a mean score of 42.7 with an SD of the mild range. The PROMIS Fatigue survey had a mean 10.0 (range 35.2–80.5), with 2.5% of the children scoring score of 45.1 with an SD of 16.4 (range 30.3–84). Fifteen in the severe depression range, 5% in the moderate range, percent of children scored in the severe fatigue range, 10% and 12.5% in the mild range. The PROMIS Anxiety sur- in the moderate range, and 7.5% in the mild range. A sum- vey had a mean score of 45.8 with an SD of 11.7 (range mary of PROMIS and PedMIDAS scores is provided in 33.5–73.0), and 2.5% of the children scored in the severe Table 2.

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TABLE 2. Summary of PROMIS and PedMIDAS scoring Measure Mean Score (SD) Median Score Min Score Max Score Scores in Mild, Moderate, & Severe Ranges, No. (%) PedMIDAS 20.2 (66.1) 0 0 386 Little/none: 32 (80); mild 2 (5); moderate 2 (5); severe 4 (10); total abnormal: 8 (20) PROMIS 42.7 (10.0) 37.8 35.2 80.5 w/in normal limits: 32 (80); mild 5 (12.5); moderate 2 (5); Depression severe 1 (2.5); total abnormal: 8 (20) PROMIS Anxiety 45.8 (11.7) 41.9 33.5 73.0 w/in normal limits: 24 (60); mild 8 (20); moderate 7 (17.5); severe 1 (2.5); total abnormal: 16 (40) PROMIS Fatigue 45.1 (16.4) 37.9 30.3 84.0 w/in normal limits: 27 (67.5); mild 3 (7.5); moderate 4 (10); severe 6 (15); total abnormal: 14 (35)

Table 3 gives a summary of the association between and whether a child had ever had a cluster of shunt fail- PedMIDAS and PROMIS scores and the clinical and ures. While one possible explanation for this finding is demographic variables. There was a statistically signifi- that shunt operations increase headache burden, it is also cant association between PedMIDAS score and having at possible that children with more headaches are more fre- least one cluster of hydrocephalus surgeries (p = 0.003). quently evaluated and surgically treated for shunt mal- When testing PROMIS measures for associations with function. It is important to note that this association is clinical and demographic factors, there were no statisti- based on a relatively small number of observations, and cally significant associations between depression, anxiety, it should be considered as hypothesis generating. Further or fatigue scores and any clinical or demographic factor. study will be required to better understand the relation- Finally, we tested for associations between anxiety, ship between chronic headache symptoms and number of depression, or fatigue, as measured by the PROMIS in- shunt surgeries. struments, and headache burden (PedMIDAS). Consider- In this sample of children with hydrocephalus, mean ing PedMIDAS as a dichotomous variable (little/none vs scores for anxiety, depression, and fatigue were all below mild/moderate/severe headache burden), headache burden the population means by approximately 0.5 SDs. Of note, was significantly associated with anxiety (p = 0.004) and the SD in our sample for each of these measures was close fatigue (p = 0.0001). There was no correlation between to the expected SD of 10 (with the exception of fatigue, headache burden and depression. These findings can be which was 16.4). This indicates that our sample behaved seen in Figs. 1–3. as expected for this instrument, with a slightly greater spread in the fatigue scores. The mean scores in this study are similar to mean scores on the PROMIS Anxiety, De- Discussion pression, and Fatigue measures found in a study of pedi- We have conducted a cross-sectional study of children atric asthma, cancer, and chronic kidney disease patients, with hydrocephalus during routine pediatric neurosur- indicating levels of psychological comorbidity in children gery clinic visits. There have been very few studies on the with hydrocephalus similar to those in other chronic con- burden of headache in children with hydrocephalus, even ditions.22 Despite reassuring mean scores for children with though these children are thought to be at high risk for hydrocephalus, there were children whose scores indicat- chronic headache complaints.18 In addition, this is the first ed above-average levels of psychological comorbidities study to quantify the severity of important psychological (20% depression, 40% anxiety, and 35% fatigue). Identify- symptoms (anxiety, depression, and fatigue) in children ing these children may provide an opportunity to improve with hydrocephalus. It is important to note that the chil- care delivery. In an attempt to determine risk factors for dren included in this study were attending routine follow- psychological comorbidities, we tested for associations up clinic visits without signs or symptoms of shunt mal- between PROMIS scores and clinical and demographic function. Therefore, these results apply to children living variables. This testing yielded no significant association with hydrocephalus as a chronic disease. between any of the tested variables and anxiety, depres- The PedMIDAS instrument was designed to measure sion, or fatigue scores. the burden of disease from headaches and has primarily Finally, we observed a significant association between been used in studies of children with migraine or oth- headache burden and two psychological variables: anxi- er types of headache. In the present study, we used the ety and fatigue. This analysis showed correlation only, so PedMIDAS as a screening tool for chronic headache in it may be that children who are more anxious then have children with hydrocephalus. There is precedent for us- more difficulty with headache or that a worse headache ing the PedMIDAS in this way.19–21 In our sample, 20% of contributes to anxiety and fatigue. Further study will be the children reported mild, moderate, or severe disability required to explore these relationships in more depth. from headache symptoms. Our findings are similar to the However, this association reinforces the importance of burden of headache-related disability found in studies of attention to psychological factors in attempts to improve children without chronic disease.19–21 overall well-being in these children. We observed a correlation between headache burden In summary, we found a burden of headache, anxiety,

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TABLE 3. Score associations with clinical and demographic variables Spearman Correlation or Median Score (min, max) Variable Depression Anxiety Fatigue PedMIDAS Age (n = 39) 0.059 (p = 0.719) −0.108 (p = 0.5137) 0.029 (p = 0.862) 0.128 (p = 0.437) Gestational age at birth (n = 32) 0.329 (p = 0.066) 0.09 (p = 0.625) 0.092 (p = 0.618) 0.092 (p = 0.618) No. of procedures (n = 37) −0.051 (p = 0.763) 0.22 (p = 0.191) −0.110 (p = 0.516) 0.277 (p = 0.097) Distance from hospital (n = 36) 0.215 (p = 0.2077) 0.292 (p = 0.084) 0.128 (p = 0.457) 0.024 (p = 0.890) Sex p = 0.896 p = 0.426 p = 0.039 p = 0.26 Male (n = 20) 37.9 (35.2, 56.1) 41.3 (33.5, 70.4) 34.7 (30.3, 67.1) 0.0 (0.0, 386) Female (n = 20) 37.8 (35.2, 80.5) 45.0 (33.5, 64.8) 47.8 (30.3, 84.0) 0.0 (0.0, 12) Race p = 0.8 p = 0.81 p = 0.943 p = 0.687 White (n = 25) 40.6 (35.2, 53.9) 42.1 (33.5, 70.4) 41.3 (30.3, 77.8) 0.0 (0.0, 386) Non-white (n = 15) 35.2 (35.2, 80.5) 41.7 (33.5, 62.9) 35.1 (30.3, 84.0) 0.0 (0.0, 50) Caregiver age in yrs p = 0.664 p = 0.794 p = 0.843 p = 0.758 21 & over (n = 33) 40.4 (35.2, 80.5) 41.7 (33.5, 70.4) 41.3 (30.3, 77.8) 0.0 (0.0, 386) Below 21 (n = 5) 41.4 (35.2, 64.5) 46.4 (33.5, 53.5) 35.1 (30.3, 84) 0.0 (0.0, 54) Caregiver education p = 0.142 p = 0.536 p = 0.721 p = 0.653 College/grad school (n = 18) 45.6 (35.2, 56.1) 44.4 (33.5, 70.4) 43.4 (30.3, 77.8) 0.0 (0.0, 386) Other (n = 20) 35.2 (35.2, 80.5) 42.1 (33.5, 64.8) 35.1 (30.3, 84.0) 0.0 (0.0, 170) Marital status p = 0.8 p = 0.917 p = 0.612 p = 0.06 Married/partnered (n = 21) 41.3 (35.2, 64.5) 41.7 (33.5, 70.4) 37.8 (30.3, 72.0) 0.0 (0.0, 170) Other (n = 17) 35.2 (35.2, 80.5) 43.3 (33.5, 62.9) 45.4 (30.3, 84.0) 3.0 (0.0, 386) Child insurance status p = 0.132 p = 0.489 p = 0.329 p = 0.304 Private (n = 17) 48.7 (35.2, 64.5) 46.4 (33.5, 70.4) 35.1 (30.3, 77.8) 0.0 (0.0, 386) Other (n = 20) 35.2 (35.2, 80.5) 41.3 (33.5, 64.8) 41.9 (30.3, 84.0) 1.0 (0.0, 170) ETV alone p = 0.601 p = 0.972 p = 0.777 p = 0.7 Yes (n = 2) 37.8 (35.2, 40.4) 46.0 (33.5, 58.6) 51.15 (30.3, 72.0) 15.5 (0.0, 31) No/none (n = 34) 35.2 (35.2, 80.5) 41.3 (33.5, 70.4) 36.45 (30.3, 84.0) 0.0 (0.0, 386) No. of clusters p = 0.811 p = 0.123 p = 0.281 p = 0.003 0 (n = 29) 35.2 (35.2, 80.5) 38.8 (33.5, 70.4) 35.1 (30.3, 84.0) 0.0 (0.0, 50) At least 1 (n = 6) 35.2 (35.2, 51.6) 51.0 (33.5, 64.8) 51.7 (30.3, 77.8) 47 (0.0, 386) Shunt infection p = 0.213 p = 0.15 p = 0.579 p = 0.303 None (n = 30) 35.2 (35.2, 56.1) 39.9 (33.5, 70.4) 36.5 (30.3, 84.0) 0.0 (0.0, 170) At least 1 (n = 7) 46.8 (35.2, 80.5) 50.9 (33.5, 62.9) 42.5 (30.3, 77.8) 2.0 (0.0, 386) No. of EVDs p = 0.942 p = 0.644 p = 0.99 p = 0.293 None (n = 24) 37.8 (35.2, 56.1) 39.9 (33.5, 70.4) 36.5 (30.3, 84.0) 0.0 (0.0, 170) At least 1 (n = 12) 35.2 (35.2, 80.5) 45.0 (3.5, 62.9) 38.5 (30.3, 77.8) 1.0 (0.0, 386) Etiology of HCP p = 0.1 p = 0.291 p = 0.633 p = 0.551 (n = 1) 35.2 (NA) 33.5 (NA) 30.3 (NA) 0 (NA) IVH (n = 8) 35.2 (35.2, 49.4) 39.0 (33.5, 64.8) 40.2 (30.3, 68.3) 0.0 (0.0, 170) MMC (n = 8) 48.2 (35.2, 80.5) 49.4 (33.5, 62.9) 43.4 (30.3, 64.6) 2.0 (0.0, 18) Other (n = 20) 35.2 (35.2, 51.6) 39.8 (33.5, 70.4) 35.0 (30.3, 84.0) 0.0 (0.0, 386) NA = not applicable. Boldface type indicates statistical significance. depression, and fatigue in a sample of children with hy- hydrocephalus has never known life without the disor- drocephalus that is similar to the burden in the general der. While their surgeons see the burden of their disease, population or other samples of children without chronic it may be that they are well adjusted to life with hydro- disease. We hypothesized that these children would have cephalus and are doing reasonably well. Nevertheless, as a higher burden of headache and psychological comor- in any pediatric , there may be benefit to bidities. Most children with hydrocephalus are diagnosed screening for psychological comorbidities, so help can be and first treated shortly after birth. Therefore, a child with provided where it is needed.

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FIG. 1. Boxplot of PROMIS Anxiety scores by PedMIDAS categorized FIG. 3. Boxplot of PROMIS Fatigue scores by PedMIDAS categorized scores (little/none vs mild/moderate/severe). scores (little/none vs mild/moderate/severe).

Study Limitations tients declined to participate because of time constraints, as This study has a number of limitations. The sample size enrollment in the study and completion of study measures was small, representing less than 10% of the children who frequently required staying additional time after the clinic are followed and treated for hydrocephalus at our institu- visit had concluded. Nevertheless, the sampling method tion. Patients were approached during a routine clinic visit, should have produced a relatively representative sample, and survey completion required on average 30 minutes. Pa- and the demographic makeup of the sample was similar to that of the clinic as a whole. Moreover, this was a single- institution study. While the findings are likely generaliz- able to patients in our region, they may not be applicable nationally or worldwide. Further, participation in this study was voluntary; therefore, it is possible that those who chose to participate are more differentially affected than those who did not. Since all outcomes were self-reported, recall bias may be present. The PedMIDAS questionnaire asks a number of questions about headache leading to missed days of school in the last month. Therefore, administration in the summer months, when there is no school, may result in falsely low scores. Some of our sample was collected in the summer months and is therefore a limitation of our data set. Conclusions In this study, we performed the first formal evalua- tion of the burden of headache, anxiety, depression, and fatigue on children with hydrocephalus. On average, chil- dren with hydrocephalus do not have a greater headache or psychological symptom burden than their healthy peers. A higher burden of headache may be associated with hav- ing a cluster of shunt operations. Anxiety, depression, and fatigue were not significantly associated with any clinical or demographic variable. A screening program for psy- chological comorbidity may be warranted during routine FIG. 2. Boxplot of PROMIS Depression scores by PedMIDAS catego- hydrocephalus care to identify children who may benefit rized scores (little/none vs mild/moderate/severe). from additional mental health evaluation.

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