Rosenthal NA; Bezar E; Mann S; Bachrach LK; Banerjee S; Geffner ME; Gottschalk M; Shapira SK; Hasegawa L; Feuchtbaum L

Special Article - Hypothyroidism

Annals Thyroid Res. 2017; 3(1): 95-101.

Primary Care Provider Management of Congenital Hypothyroidism Identified Through Newborn Screening

Rosenthal NA¹, Bezar E², Mann S³, Bachrach LK⁴, Banerjee S⁵, Geffner ME⁶, Gottschalk M⁷, Shapira SK⁸, Hasegawa L³ and Feuchtbaum L¹*

¹California Department of Public Health, Genetic Disease Screening Program, USA

²Public Health Foundation Enterprises, USA

³Hawai’i Department of Health Genetics Program, USA

⁴Lucile Salter Packard Children’s Hospital, Stanford School of Medicine, USA

⁵Valley Children’s Hospital, USA

⁶Children’s Hospital Los Angeles, USA

⁷UCSD School of Medicine, USA

⁸Centers for Disease Control and Prevention, USA

*Corresponding author: Lisa Feuchtbaum, California Department of Public Health, Genetic Disease Screening Program, Richmond CA 94804, Dr. PH, MPH. 850 Marina Bay Parkway, F-175, Richmond, CA 94804, USA

Received: March 17, 2017; Accepted: April 03, 2017; Published: April 18, 2017

Abstract

Objective: To assess Primary Congenital Hypothyroidism (CH) management patterns and feasibility of providing long-term care for patients with CH identified through newborn screening by Primary Care Providers (PCPs) in California and Hawaii.

Study Design: A survey was mailed to all physicians (N=823) listed as the referral doctor for confirmed patients with CH identified through newborn screening programs in both states between 01/01/2009–12/31/2013. Information was collected on CH management patterns, barriers to providing care, and knowledge on CH treatment. Descriptive statistics and bivariate logistic regression results were reported.

Results: 206 PCPs completed the survey. Among these, 78% currently have patients with CH and 91% indicated willingness to provide long-term care to new patients with CH. Among PCPs currently caring for patients with CH, 17% managed CH by themselves with limited assistance from endocrinologists; 63% were involved in managing CH but endocrinologists played a larger role than PCPs; 19% were not involved in CH care. Only 49% of PCPs correctly answered questions regarding recommended follow-up frequencies and 23% knew the correct age for a trial off levothyroxine for suspected transient CH. Top two perceived barriers to providing long-term care included “need guidance or support from endocrinologists” (61%) and “not familiar with CH treatment guidelines” (28%).

Conclusion: The majority of PCPs surveyed are willing to provide long-term care to patients with CH, but need support from endocrinologists and increased knowledge about current treatment guidelines.

Keywords: Congenital hypothyroidism; Primary care; Management; Newborn screening

Abbreviations

CH: Primary Congenital Hypothyroidism; NBS: Newborn Screening; LTFU: Long-Term Care and Follow-Up; PCP: Primary Care Providers; AAP: American Academy of Pediatrics; CME: Continuing Medical Education

Introduction

Primary Congenital Hypothyroidism (CH) is the most common preventable cause of neurocognitive disability (mental retardation) [1] and is the most common disorder diagnosed through Newborn Screening (NBS) [2]. CH affects about 1 in 2000 California newborns and 1 in 3000 Hawaii newborns, with over 245 new cases diagnosed in both states each year [2,3] CH refers to thyroid hormone deficiency due to dysfunction of the thyroid gland. About 80%?85% of permanent CH cases are caused by developmental failure or maldevelopment of the thyroid gland (i.e., aplasia, hypoplasia, or ectopia). Other permanent cases are caused by an inherited enzyme deficiency affecting synthesis of thyroid hormone (dyshormonogenesis) or a thyrotropin receptor defect [1,4]. Although early detection and initiation of treatment within the newborn period have nearly eradicated severe mental disabilities among patients with CH in countries with NBS, unfavorable clinical outcomes are still present in a subset of patients [5]. CH severity and treatment inadequacy have been associated with a lower Intellectual Quotient (IQ) [6-9], lower educational attainment [10-12], and sensorineural hearing loss [10,13,14]. Patients with severe or inadequately treated CH also have increased risk of congenital malformations (e.g., anomalies of cardiac and nervous system and eyes), obesity [10,15-18], and impaired growth, puberty, and fecundity [19-22]. These findings highlight the importance of adequate treatment and monitoring to maintain optimal thyroid function throughout life to ensure the best neuro developmental, physical, and social outcomes in patients with CH.

Specific diagnostic, treatment, and follow-up guidelines for children with CH have been published by both the American Academy of Pediatrics (AAP) and the European Society for Paediatric Endocrinology [23,24]. Both guidelines recommend frequent followups during childhood. However, a U.S. study using both public and private health insurance data found that over one-third of patients with CH discontinued treatment by three years of age [25]. Although the precise incidence of transient hypothyroidism is unknown (recent estimates range from 13.5%to 54.5% [26-28]), the authors of the analysis suggested that most cases of treatment discontinuation were likely not transient, but parent-initiated without physician supervision [23,25,27]. A follow-up study of patients with CH detected by the Michigan NBS Program showed that 25% of the patients were no longer being treated by three years of age, among which 83% had stopped treatment without medical supervision [29]. Of those who underwent a medically-supervised trial off medication, treatment was resumed in 87% of the cases [29]. These data suggest that discontinuation of treatment without medical supervision is a problem that could result in suboptimal outcomes for many patients. Due to the paucity of long-term follow-up data from the United States, it is unclear how patients with CH are being managed as they age and the potential adverse effects of inadequate treatment or premature discontinuation of levothyroxine treatment.

In both California and Hawaii, patients who screen positive for CH in state NBS programs are usually referred by their Primary Care Providers (PCPs) to endocrinologists for further testing and confirmatory diagnosis. In California, a large percent of screen positive patients are referred to state-contracted endocrine centers to provide follow-up care. However, feedback from endocrinologists indicated that these centers usually do not have the resources needed to follow every patient with CH as frequently as recommended by the AAP guidelines, especially during the first three years of life, when treatment adherence is most critical. Furthermore, it is often more practical and convenient for patients with CH to receive care from their PCPs who are usually in closer proximity and more readily available, especially in rural areas where fewer endocrinologists are available [30]. Parks et al found that in Texas during 2004-2006, 64% of patients with CH were followed and reported by endocrinologists in the first year of life and the proportion decreased to 26% in the second year of life. Underreporting from endocrinologists might exist, but these findings indicated that some patients might be followed by primary care pediatricians [31]. The AAP-recommended CH follow-up visits can be completed during routine well-baby visits. Endocrinologists can serve as consultants after the initial evaluation of the case is completed and provide guidance about modifying treatment when questions arise. The consensus is that PCPs should take more responsibility for providing long-term care and encouraging adherence to treatment for patients with CH with the support and consultation from pediatric endocrinologists.

These recommendations are consistent with the patient-centered medical home model for providing primary care that has been widely endorsed by physicians’ associations, including the AAP and the American Academy of Family Physicians [32]. Studies have shown that, for children and youth with special health-care needs, care provided through a medical home is associated with better care coordination, higher satisfaction with care, and fewer emergency room visits or hospitalizations [33-36]. Compared to metabolic disorders screened for by NBS, CH is easier to manage since it primarily involves the administration of a single drug. Thus, with proper training, the locus of the medical home can be with the PCP when that provider is supported by apediatric endocrinologist for challenging and more nuanced cases. Increased PCP involvement and “ownership” of care for patients with CH would likely improve quality of care, disease management and health outcomes. Understanding the hurdles impeding management of children with CH by pediatricians and whether it is feasible to shift the Long-Term Follow-Up (LTFU) responsibility to PCPs with support from pediatric endocrinologists will strengthen the medical home for these patients.

The purpose of this study was four-fold: 1) to evaluate the current CH case management patterns; 2) to assess the willingness and capability of PCPs to provide LTFU for patients with CH; 3) to identify potential barriers for PCPs to provide LTFU to patients with CH; and 4) to assess PCPs’ willingness to obtain informed consent and provide data to an existing database to evaluate quality of care and patient outcomes for possible research endeavors in the future.

Methods

A cross-sectional survey of PCPs was conducted from February through June of 2014. All PCPs who were listed as the referral doctors for at least one patient with CH born between 2009-2013 were selected from the California and Hawaii NBS databases. A total of 801 physicians from California and 22 physicians from Hawaii were invited to participate in the study.

The survey included five key components relevant to PCPs: 1) current practice in managing patients with CH; 2) barriers and resources needed to conduct CH LTFU; 3) willingness and capability to conduct CH LTFU; 4) willingness and capability to obtain informed consent from patients and provide data to a LTFU database; and 5) clinical outcomes of patients with CH being seen by the PCPs. Results of the fifth component will be reported in a future paper. To maximize response rate, we included a personalized cover letter in the surveymailing packet that described the significance and objectives of the study. To better understand the reasons for non-response, we asked each doctor who was unable to complete the questionnaire to indicate reasons for non-response on a separate “Non-Response Card” enclosed with the initial mailing. A gift card incentive was offered to PCPs who completed either the full survey or the non-response card. An on-line version of the same survey was also made available at the same time. One hundred and thirteen mailing packets were returned as undeliverable after the initial mailing. We were able to obtain their most up-to-date addresses through multiple modes (e.g., internet search and calling the listed offices) for 87 doctors and resent the packets. We failed to find usable addresses for 26 doctors and they were excluded from the response rate calculation. Additionally, we sent out two reminder letters to non-responding PCPs, one and two months after the initial mailing, to boost the response rate.

All data were analyzed using SAS version 9.3 for Windows (SAS Institute, Cary, NC). Continuous variables were expressed as medians with range and categorical variables were presented as numbers and percentages. Descriptive statistics were calculated and the differences between the two states were assessed using Chi-square and Fisher’s exact tests for categorical variables and Wilcoxon Signed Rank Test for continuous variables to compare the difference in medians. Bivariate logistic regression modeling was used to assess the association between selected covariates and key outcome variables. Crude Odds Ratios (ORs) and their 95% Confidence Intervals (CIs) were reported. This study was approved by the State of California, Health and Human Services Agency, Committee for the Protection of Human Subjects, Project Number 13-08-1317.

Results

Response rates and demographics

A total of 238 doctors in California and 10 doctors in Hawaii responded to the mailing, among whom 226 completed the survey (128 paper surveys and 98 on-line surveys) and 19 returned the Non- Response Card (Figure 1). Among doctors who completed the survey, 20 were not PCPs and their responses were excluded from the final analysis. The total response rate was 28% for California doctors and 45% for Hawaii doctors. Among the 19 doctors/offices that returned the Non-Response Card and indicated a reason for not completing the survey, four did not have time to do the survey, seven were specialists instead of PCPs, six did not have patients with CH, one doctor was deceased, and one had security concerns.

Figure 1: Response rate of the Primary Care Providers (PCPs) surveyed in California (CA) and Hawaii (HI) in 2014.

    
    
    Figure 1: Response rate of the Primary Care Providers (PCPs) surveyed in
California (CA) and Hawaii (HI) in 2014.

Among the 206 PCPs who completed the survey, 51% were male, 46% were White, 39% were Asian, 7% were of Hispanic origins and 8% were of other race or ethnicity (Table 1). The majority of respondents were primary care pediatricians and 8% identified themselves as family physicians. Nearly 50% were in private practice, 33% were in group practice, 13% were in community health centers, and only 4% were in a hospital-based practice or HMO. Respondents had been in medical practice for a median of 18 years (range 2–43 years). There was no statistically significant difference in these variables between respondents from California and Hawaii.

Table 1: Characteristics of the responding primary care providers in California (CA) and Hawaii (HI).




  
    Characteristics 
    CA    (%, n=196) 
    HI (%,    n=10) 
    All    (%, n=206) 
  
  
    Male    sex 
    51
    60
    51
  
  
    Race/ethnicity 
    
    
    
  
  
    Hispanic/Latino
    7
    0
    7
  
  
    White
    47
    20
    46
  
  
    Asian
    38
    60
    39
  
  
    Black
    3
    0
    3
  
  
    Mixed    or Other
    5
    20
    5
  
  
    Medical    specialty 
     
    
    
  
  
    Family    practice
    9
    0
    8
  
  
    Pediatrics
    91
    100
    92
  
  
    Type    of practice 
    
    
    
  
  
    Private    practice
    45
    50
    45
  
  
    Group    practice
    33
    30
    33
  
  
    Hospital-based    practice
    5
    0
    4
  
  
    HMO
    4
    0
    4
  
  
    Community    health center
    13
    20
    13
  
  
    Years    in medical practice 
    
    
    
  
  
    Median    (range)
    18 (2, 43)
    19 (8, 29)
    18 (2, 43)
  
  
    Note: There was no statistically    significant difference between CA and HI for any of the above variables.



Table 1:  Characteristics of the responding primary care providers in California
(CA) and Hawaii (HI).

Case management patterns and long-term care feasibility

When California PCPs were asked who usually manages CH for their patients, 17% indicated that their patients’ CH was mainly managed by themselves, but endocrinologists were involved; 63% were involved, but patients’ CH was mainly managed by endocrinologists; and 19% indicated that their patients’ CH was solely managed by endocrinologists. Among Hawaii PCPs, 75% indicated that their patients’ CH was mainly managed by endocrinologists, but they were also involved and the rest indicated that their patients’ CH was solely managed by endocrinologists. Among all respondents, “need guidance or support from endocrinologists” and “not familiar with the CH treatment guidelines” were listed as the two most commonly perceived barriers for PCPs to provide long-term care for patients with CH.

Over 90% of responding PCPs in both states indicated willingness to provide long-term care for new patients with CH. Compared to PCPs who mainly manage patients’ CH by themselves, those who were not involved in their patients’ CH management had 85% lower odds of being willing to provide long-term care for new patients with CH (OR=0.15, 95% CI: 0.03, 0.77). Type of practice and currently having patients with CH were not associated with willingness to provide long-term care for new patients with CH (Table 2 and 4).

Table 2: Case management patterns, perceived barriers, and willingness to providing Long-Term care and Follow-Up (LTFU) for patients with primary Congenital Hypothyroidism (CH).




  
    Variables
    CA (%) 
    HI (%) 
    All (%) 
  
  
    Who manages patients’ CH conditions 
    
    
    
  
  
    Solely by PCPs
    1
    0
    1
  
  
    Mainly by PCPs, but endocrinologists are involved
    17
    0
    16
  
  
    Mainly by endocrinologists, but PCPs are involved
    63
    75
    64
  
  
    Solely by endocrinologists
    19
    25
    19
  
  
    Willing to provide long-term care for new patients with CH 
    
    
  
  
    Yes
    66
    80
    67
  
  
    Maybe
    25
    20
    24
  
  
    No
    6
    0
    6
  
  
    Don't know
    3
    0
    3
  
  
     
      Perceived barriers for providing long-term care 
    
    
    
  
  
    Need guidance or support from endocrinologists
    60
    80
    61
  
  
    Not familiar with the CH treatment guidelines
    28
    40
    28
  
  
    Need more staff time to coordinate care
    15
    10
    15
  
  
    Patients are not compliant with care
    14
    10
    14
  
  
    Don’t have enough time
    12
    10
    12
  
  
    CH long-term care is too complicated
    11
    0
    10
  
  
    Don’t get enough reimbursement
    11
    0
    10
  
  
    Anticipate no barriers
    20
    10
    20
  
  
     
      Willingness to obtain informed consent 
     
      75
     
      100
     
      76
  
  
     
      Difficulties in obtaining informed consent 
    
    
    
  
  
    Very difficult
    4
    0
    4
  
  
    Difficult, but doable
    35
    20
    34
  
  
    Not difficult at all
    39
    70
    40
  
  
    Don't know
    22
    10
    21
  
  
     
      Willingness to provide LTFU data 
    
    
    
  
  
    Yes
    67
    90
    68
  
  
    Maybe
    19
    10
    19
  
  
    No
    3
    0
    3
  
  
    Don't know
    11
    0
    10
  
  
     
      Reasons for not willing to provide LTFU data 
    
    
    
  
  
    Don’t have enough staff or time to enter data
    37
    0
    37
  
  
    Do not provide long-term care for patients’ CH
    37
    0
    37
  
  
    Have concerns over patient’s privacy
    19
    0
    19
  
  
    The LTFU data is not relevant to clinical practice
    7
    0
    7
  
  
    It is not important to collect LTFU data for CH
    0
    0
    0
  
  
    Compensation required to provide LTFU data
    
    
    
  
  
    $50/patient/year 
    5
    20
    6
  
  
    $100/patient/year
    18
    20
    18
  
  
    $150/patient/year
    14
    0
    13
  
  
    $200/patient/year
    18
    0
    17
  
  
    No compensation is needed
    18
    40
    19
  
  
    Don't know/Other
    27
    20
    27
  
  
    Note: There    was no statistically significant difference between California and Hawaii for    any of the above variables. CA: California; HI: Hawaii.



Table 2:  Case management patterns, perceived barriers, and willingness to
providing Long-Term care and Follow-Up (LTFU) for patients with primary
Congenital Hypothyroidism (CH).

Over two-thirds of PCPs in both states expressed willingness to obtain informed consent from patients to record and share LTFU data on their patients with CH. Nearly 75% of PCPs consider obtaining informed consent doable with little or no difficulties. About 87% of PCPs from both states indicated willingness to report LTFU data if given a reasonable compensation. PCPs currently caring for patients with CH were more likely to indicate willingness to provide LTFU data than those who had no patient. Among the 27 California PCPs who were not willing to collect LTFU data, over one-third indicated “do not have enough staff or time to enter data” or “do not provide long-term care for patients’ CH condition” as the main reasons for their unwillingness to do so. In terms of compensation required to provide LTFU data, 73% of PCPs would be satisfied with a compensation less than $200 per patient per year (Tables 2 and 4).

Knowledge about CH-related management

When asked about the recommended frequency of blood tests for patients with CH in three different age groups (<6 months, 6 months to 3 years, and >3 years), the proportion of PCPs who correctly answered each question was72%, 60%, and 73% for each age group, respectively. Only 49% of PCPs correctly answered all three questions about the recommended follow-up frequencies (Table 3). There was no statistical difference in the prevalence of knowing the recommended follow-up frequencies between PCPs who reported different management patterns, type of practice, or number of patients (Table 4).

Table 3: Knowledge of current treatment guidelines for primary congenital hypothyroidism.




  
    Variables 
    CA (%) 
    HI (%) 
    All (%) 
  
  
    Know the recommended frequency of blood tests 
    
    
    
  
  
    In the first six months of age
    72
    70
    72
  
  
    Between six months and three years of age
    59
    70
    60
  
  
    Over three years of age
    74
    50
    73
  
  
    Know all frequencies
    49
    50
    49
  
  
     
      Familiarity with indications for trial off levothyroxine 
    
    
    
  
  
    Very familiar
    3
    0
    2
  
  
    Somewhat familiar
    29
    20
    28
  
  
    Not familiar
    54
    70
    55
  
  
    Do not know
    15
    10
    14
  
  
     
      Know at what age for trial off levothyroxine 
     
      23
     
      30
     
      23
  
  
    Note: There    was no statistically significant difference between California and Hawaii for    any of the above variables. CA: California; HI: Hawaii.



Table 3:  Knowledge of current treatment guidelines for primary congenital
hypothyroidism.

Table 4: Association between selected covariates and key outcome variables.




  
    Covariates 
    Know recommended follow-up frequencies 
      OR (95% CI)* 
    Willing to provide care for new patients with CH^ (Yes and    Maybe) 
      OR (95% CI)* 
    Willing to provide LTFU data (Yes and Maybe) 
      OR (95% CI)* 
    Likelihood to participate in CME (Very likely and Likely) 
      OR (95% CI)* 
  
  
    Currently having =1 patients with CH^
    
    
    
  
  
    Yes
    1.1 (0.5, 2.1)
    1.8 (0.6, 5.4)
    3.1 (1.3, 7.6)
    3.0 (1.1, 8.5)
  
  
    No
    Reference
    Reference
    Reference
    Reference
  
  
    Management patterns 
    
    
    
    
  
  
    Mainly by PCP^$, but endocrinologists are    involved
    Reference
    Reference
    Reference
    Reference
  
  
    Mainly by endocrinologists, but PCP is involved
    0.9 (0.4, 2.0)
    4.1 (0.5, 30.6)
    1.3 (0.3, 5.2)
    3.0 (0.8, 11.4)
  
  
    Solely by endocrinologists
    1.2 (0.4, 3.2)
    0.15 (0.03, 0.77)
    0.6 (0.1, 3.0)
    1.2 (0.3, 5.3)
  
  
    Type of practice 
    
    
    
    
  
  
    Private practice
    Reference
    Reference
    Reference
    Reference
  
  
    Group practice
    1.1 (0.6, 2.0)
    1.1 (0.4, 3.0)
    1.6 (0.5, 4.8)
    0.4 (0.1, 1.2)
  
  
    Hospital-based practice
    0.12 (0.01, 1.04)
    1.0 (0.1, 8.7)
    0.25 (0.05, 1.14)
    0.22 (0.04, 1.35)
  
  
    HMO^$
    1.7 (0.4, 7.4)
    N/A
    0.12 (0.03, 0.57)
    0.22 (0.04, 1.35)
  
  
    Community health center
    1.7 (0.7, 4.1)
    3.2 (0.4, 26.3)
    0.5 (0.2, 1.7)
    1.8 (0.2, 15.5)
  
  
    *OR: Odds Ratio; CI: Confidence    Interval; ^CH: Primary Congenital Hypothyroidism
      Significant odds ratios are shown in bold. ^$PCP:    Primary Care Providers; HMO: Health Maintenance Organization.



Table 4:  Association between selected covariates and key outcome variables.

When assessing PCPs’ familiarity with indications for a trial off levothyroxine therapy to assess the transient status of CH, only 2% were very familiar, 28% were somewhat familiar, 55% were not familiar, and 14% did not know. Only 23% of PCPs in both states knew the recommended age for such a trial (Table 3).

Preference for CH-related continuing medical education

Over 80% of responding PCPs in both states indicated being “likely” or “very likely” to participate in Continuing Medical Education (CME) about CH if such courses are available. PCPs who reported currently having patients with CH were more likely to participate in CH-related CME than those without patients (Table 4). With regard to the preferred format for CME, over 60% of respondents chose webinars, about one-third chose in-person classes, and one-third chose grand rounds presentations.

Discussion

This study of current management patterns of patients with CH and feasibility of PCP involvement in long-term care provides important insights on how to assure high quality primary care and optimal clinical outcomes for patients with CH. Although the majority of PCPs who completed the survey were willing to provide long-term care to their patients with CH, endocrinologists were identified as assuming the primary management responsibilities in most of the cases in both states. The two most commonly perceived barriers by PCPs to providing long-term care for patients with CH were: needing guidance from endocrinologists and lack of familiarity with the current CH treatment guidelines. We identified a general lack of knowledge about CH treatment and management among responding PCPs. Nonetheless, the majority of respondents were willing to provide long-term care for new patients with CH. Specifically, they considered it feasible to obtain informed consent from patients with CH or their guardians, and would be willing to provide LTFU data to an existing database for a compensation of $200 or less per patient per year.

Long-term follow-up data for patients identified through NBS can help evaluate the clinical outcomes and health service utilization patterns of patients with diagnosed disorders in diverse populations. Multiple national agencies have released initiatives to establish a comprehensive, sustainable, and feasible long-term follow-up data collection system to improve the ultimate health outcomes of patients with screened disorders [37-40]. For patients with CH, optimal treatment throughout life is critical for achieving the best neurological, physical, economic, and social outcomes [24,40]. How to effectively deliver the needed care to all patients with CH and ensure adequate treatment for everyone remains a challenge in both California and Hawaii. National data showed that there is a shortage of pediatric endocrinologists nationwide; the average time that patients have to wait to see a pediatric endocrinologist is 10.3 weeks and the average distance to pediatric endocrinology care is over 26 miles [30]. Findings from this study indicate the possibility of integrating such care into primary care practices. The majority of PCPs in both states were willing to take the responsibility of caring for patients with CH with support and guidance from endocrinologists. Future efforts should be focused on how to improve the communication between PCPs and endocrinologists and to ensure that each patient receives appropriate and comprehensive care.

To our knowledge, this is the first survey of PCPs to identify a knowledge gap in CH management. To increase PCPs’ capability for providing long-term care for patients with CH, one avenue would be to provide CME opportunities for PCPs to learn about current guidelines for CH treatment and management. In response to the documented strong interests in CME training from responding PCPs, we have since developed a CH management curriculum entitled, “Congenital Hypothyroidism: What Every Pediatrician Needs to Know.” This course has been offered through Pediatric Grand Rounds throughout California and Hawaii. Building off of this CME course for PCH providers, Stanford University recently developed an online CME course on CH that is applicable more broadly to pediatric care providers (https://med.stanford.edu/cme/courses/ online/hypothyroidism.html). These teaching modules review how to confirm diagnosis, initiate treatment if required, monitor thyroid function, adjust levothyroxine dosing for infants, children and adolescents, and underscore the need for providers to educate families about the importance of adherence to treatment. We hope that such efforts will increase the confidence and competence of PCPs to manage patients with CH and improve their health outcomes.

We recognize a key limitation in this study. The overall response rate of completed surveys was 28.4%, despite the substantial efforts to improve the response rate as outlined in methods section [41]. The top three reported reasons for not completing the survey among those who returned the Non-Response Card were: being specialists themselves, having no time, and not currently having any patient with CH. The first two reasons may not cause any response bias. The third reason, not currently caring for any patient with CH, could result in an overestimation of several outcome variables including the percentage of doctors currently having patients with CH and knowledge about CH management. It is possible that PCPs currently caring for patients with CH would be more likely to participate in the study and have better knowledge than those without any patient with CH. Prior studies have shown that provider surveys are resistant to non-response bias and our response rate was better than other provider surveys of similar length [41-44]. We believe that the findings may be reasonably representative of PCPs in both states.

Conclusion

PCPs in both California and Hawaii expressed willingness to provide long-term care for patients with CH, but to fulfill the responsibility they need additional training on up-to-date CH-related treatment and management guidelines, as well as strong support from endocrinologists who can provide consultation for dealing with difficult cases. In addition, PCPs stated willingness to provide longterm follow-up data for patients with CH to existing databases. In future efforts to improve the clinical outcomes for patients with CH or other disorders identified by NBS and ensure that they receive high quality primary care services we should try to better engage PCPs in the care process and improve the collaboration between PCPs and specialists. More research is needed to assess the effectiveness of educational efforts for PCPs and the feasibility of conducting longterm follow-up for rare diseases in primary care settings.

Acknowledgement

We would like to thank the Human Resources and Services Administration for providing funding for this study. We also thank all doctors who participated in the survey, and colleagues at the California Genetic Disease Screening Laboratories and Genetic Disease Screening Program, including, Tracey Bishop, Shellye Lessing, Norah Ojeda, Bob Currier, Sona Saha and Hari Haran,for their help with the survey contents and administration. Drs. Ning Rosenthal and Lisa Feuchtbaum had full access to all the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. This project was supported by the Health Resources and Services Administration (HRSA) of the U.S. Department of Health and Human Services (HHS) under grant D93MC26187-02-00: Demonstration Projects for Integrating Newborn Screening Long Term Follow-Up into Primary Care Practice. This information or content and conclusions are those of the authors and should not be construed as the official position or policy of, nor should any endorsements be inferred by HRSA, the Centers for Disease Control and Prevention, HHS or the U.S. Government. None of the coauthors has conflicts of interest relevant to this article to disclose.

Funding Source

This project is supported by the Health Resources and Services Administration (HRSA) of the U.S. Department of Health and Human Services (HHS) under grant D93MC26187-02-00: Demonstration Projects for Integrating Newborn Screening Long Term Follow- Up into Primary Care Practice. The funding agency does not have a conflict of interest with this research or this manuscript and did not influence (1) the study design; (2) the collection, analysis, and interpretation of data; (3) the writing of the report; and (4) the decision to submit the paper for publication.

This information or content and conclusions are those of the authors and should not be construed as the official position or policy of, nor should any endorsements be inferred by HRSA, the Centers for Disease Control and Prevention, HHS or the U.S. Government.

References

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Citation: Rosenthal NA, Bezar E, Mann S, Bachrach LK, Banerjee S, Geffner ME, et al. Primary Care Provider Management of Congenital Hypothyroidism Identified Through Newborn Screening. Annals Thyroid Res. 2017; 3(1): 95-101.

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Characteristics	CA (%, n=196)	HI (%, n=10)	All (%, n=206)
Male sex	51	60	51
Race/ethnicity
Hispanic/Latino	7	0	7
White	47	20	46
Asian	38	60	39
Black	3	0	3
Mixed or Other	5	20	5
Medical specialty
Family practice	9	0	8
Pediatrics	91	100	92
Type of practice
Private practice	45	50	45
Group practice	33	30	33
Hospital-based practice	5	0	4
HMO	4	0	4
Community health center	13	20	13
Years in medical practice
Median (range)	18 (2, 43)	19 (8, 29)	18 (2, 43)
Note: There was no statistically significant difference between CA and HI for any of the above variables.