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ORIGINAL ARTICLE
Year : 2017  |  Volume : 30  |  Issue : 1  |  Page : 39-43

Impact of compliance to therapy of congenital hypothyroidism on the neurodevelopmental outcome in Sharkia Governorate


1 Department of Family Medicine, Faculty of Medicine, Menoufia University, Egypt
2 Department of Internal Medicine, Faculty of Medicine, Menoufia University, Egypt
3 Department of Family Medicine, Faculty of Medicine, Zagazig University, Zagazig, Egypt

Date of Submission18-Oct-2016
Date of Acceptance11-Dec-2016
Date of Web Publication25-Jul-2017

Correspondence Address:
Walaa S Mohamed
Department of Family Medicine, Faculty of Medicine, Zagazig University, Zagazig, 44511
Egypt
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1110-2098.211519

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  Abstract 


Objective
The objective of this study was to evaluate the compliance to congenital hypothyroidism (CH) therapy and the effect of noncompliance on the physical and mental development.
Background
CH is the most common preventable cause of mental retardation in children. Early treatment and high compliance are important prognostic factors for mental and physical development in children with CH.
Patients and methods
A prospective study design was conducted in the Endocrinology Outpatient Clinics in Sharkia Governorate health insurance center. The participants were 120 newborns and infants with CH and their mothers (patients), and 120 age-matched volunteers of healthy infants and children and their mothers (control). Data were collected using multistructured questionnaires for determining the socioeconomic state, clinical picture, personal, family and obstetric history, and Morisky Medication Adherence Scale (MMAS 8) to assess compliance. Assessment of physical growth was carried out through anthropometric measurements and assessment of mental development was conducted using Vineland-II scale for infants below 2 years and using Stanford–Binet 5 Intelligence scales for individuals of 2 years and older. Review of the results of investigations at diagnosis and during the follow-up visits are provided.
Results
Two-thirds of the patient families were highly compliant. There was a very high significant difference between different compliance grades and between patients and control groups as regards the parameters of mental and physical development. No significant difference was found between the highly compliant cases and control.
Conclusion
Compliance to therapy in our patients and/or their family was reasonable. High compliance to treatment was an important prognostic factor for normal mental and physical development in patients with CH.

Keywords: compliance, congenital hypothyroidism, medication adherence


How to cite this article:
Farahat TM, Elshorbagy EA, Hegazy NN, Mohamed WS. Impact of compliance to therapy of congenital hypothyroidism on the neurodevelopmental outcome in Sharkia Governorate. Menoufia Med J 2017;30:39-43

How to cite this URL:
Farahat TM, Elshorbagy EA, Hegazy NN, Mohamed WS. Impact of compliance to therapy of congenital hypothyroidism on the neurodevelopmental outcome in Sharkia Governorate. Menoufia Med J [serial online] 2017 [cited 2019 Aug 19];30:39-43. Available from: http://www.mmj.eg.net/text.asp?2017/30/1/39/211519




  Introduction Top


Congenital hypothyroidism (CH) is the most common congenital endocrine disorder and one of the most common preventable causes of mental retardation in children [1].

CH is defined as thyroid hormone deficiency that is present at birth, most commonly caused by a problem with thyroid gland development or disruptions in thyroid hormone synthesis [2].

The incidence of CH was 1:3000 to 1:4000 live births in iodine-sufficient countries [3]. In Egypt the incidence of CH during the period from January 2003 to December 2011 was 1:2941 live births in Fayoum [4].

Most of the newborns with CH are asymptomatic and without newborn screening many patients remained undiagnosed at birth [5].

Since delays in the diagnosis and treatment of CH may lead to neurological sequelae including mental retardation, poor motor coordination, learning disorders, early diagnosis and treatment are critical in CH [6].

Neonatal screening allows early detection and treatment of CH, and in the prevention of mental retardation that results from lack of thyroid hormone [7].

The presence of elevated thyroid stimulating hormone (TSH) levels and a low free T4 or total T4 confirms the diagnosis of primary CH. The presence of elevated TSH with normal free T4 or total T4 is consistent with subclinical primary CH [2].

The mainstay in the treatment of CH is early diagnosis and thyroid hormone replacement. Optimal care includes diagnosis before the age 10–13 days and normalization of thyroid hormone levels by the age of 3 weeks. The goal of the therapy is to achieve normal growth and mental development [8].

Factors that influence the neurodevelopmental outcome in children with CH are the age at initiation of treatment, starting dose of levothyroxine, severity of disease, serum T4 concentrations in the first 2 years of life, and compliance to therapy [1].

The aim of this work is to evaluate the physical and mental growth and the development of target cases of CH and to assess the compliance of the target families to L thyroxin therapy.


  Patients and Methods Top


The study was approved by the ethical committee of the Faculty of Medicine, Menoufia University. This prospective study was conducted from the 1 January 2015 till the end of December, 2015 in the Endocrinology Outpatient Clinics in Sharkia Governorate health insurance center.

All newborns with congenital hypothyroidism and infants and/or children who come for treatment and follow-up in the center during 6 months of the study period were enrolled in the study as a patient group. It included 120 patients (57 men and 63 women). A similar control group was recruited from 120 participants (54 men and 66 women) of age-matched and sex-matched volunteers of healthy infants and children. The mothers of the patients were assessed to evaluate the compliance to therapy.

All the study participants were subjected to the following:

Four predesigned interview questionnaires were used for determining the socioeconomic status, clinical picture of congenital hypothyroidism at presentation, personal history and family history and MMAS 8 to assess patient compliance. Patients with MMAS 8 score of 8 were considered highly compliant; patients with a score of 6 or 7 were considered medium compliant and those with a score of less than 6 were considered of low compliance.

Assessment of the physical growth was carried out through anthropometric measurements and the measurements were referred to their Z score for age.

Assessment of the mental development was conducted using Vineland-II scale for infants below 2 years of age and using Stanford–Binet 5 Intelligence Scales for individuals of 2 years or older.

Statistical analysis

Data were checked, entered, and analyzed by using SPSS, version 19 (SPSS Inc., Chicago, IL, USA). Data were expressed as mean ± SD for quantitative variables, number, and percentage for categorical variables. c2 was used to test the association between qualitative variables. Analysis of variance (F-test) was used to compare the means of more than two samples and paired t-test was used to compare just two samples; Pearson's correlation (r) was used to detect the linear relationship between two variables. A P value of less than 0.05 was considered statistically significant.


  Results Top


In this study, the incidence of CH was 1/3195 live births in Sharkia governorate during the year2015.

Most of our patients (about two-third) were highly compliant to therapy while less than one-third of the patients were low compliant to therapy and only 11.7% of patients were medium compliant [Figure 1].
Figure 1: Frequency and percent of compliance of the patients to therapy.

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There were statistically significant differences between cases and control groups regarding Vinland adaptive score, Stanford-Binet5 IQ score, height/age and head circumflex/age Z scores (P = 0.002, 0.001, 0.000 and 0.019, respectively). However, no significant difference was found as regards weight/age Z score [Table 1]. Moreover, no significant difference was found between the highly compliant cases and control group as regards the parameters of mental and physical development [Table 2].
Table 1 Comparison of the mean±SD of the IQ scores and anthropometric Z scores between the cases and control groups (N=120)

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Table 2 Comparison of the mean±SD of IQ scores and anthropometric Z scores between the highly compliant cases and control

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There was a highly statistically significant difference between high, medium, and low compliant grades regarding the Vinland adaptive score, Stanford-Binet5 score height/age and head circumference/age Z scores (P = 0.000 for each parameter). The higher the compliance grade the better the parameters of physical and mental development [Table 3].
Table 3 Comparison between different compliant grades as regards the anthropometric Z scores and IQ scores

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There were statistical significant positive correlations between the compliance score and the Vinland adaptive and Stanford-Binet5 IQ scores, height/age, and head circumference/age Z scores. However, no statistically significant correlation was found between compliance score and the weight/age Z score [Table 4].
Table 4 Correlation between the compliance score, Z scores and IQ scores

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There was a highly statistical significant difference between high, medium, and low compliant grades regarding the mean ± SD of the TSH levels during the follow-up visits (P = 0.000). The higher the TSH level, the lower the compliance grades [Table 5]. A significant negative correlation was found between the compliance score and the TSH levels at the start of the study and during the follow-up visits [Table 3] and [Table 6].
Table 5 Comparison between different compliant grades as regards the thyroid syimulating hormone level

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Table 6 Correlation between the compliance score and thyroid stimulating hormone levels

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  Discussion Top


The mainstay in the treatment of CH is early diagnosis and thyroid hormone replacement to prevent the negative impact on the physical and mental development [8].

This study aimed to evaluate the physical and mental growth and development of the target cases of CH and to assess the compliance of the target families to therapy.

In our study the incidence of CH in Sharkia governorate was 1/3195 live births during 2015. This is similar to prevalence reported by Dabbous et al. [9] (1:2567 and 1:3306 live births in 2001 and 2002 in Alexandria, Egypt); Abdel-Rasoul et al. (1:3000 live births in Menoufiya governorate from year 2003 till 2008) [10], and Corbetta et al. [3] (1:3000 to 1:4000 live births in iodine-sufficient countries in 2009.

Concerning compliance to thyroid hormone therapy, most of our patients (about two-thirds) are highly compliant to therapy, while less than one-third of the patients are low compliant. This is consistent with data recorded by Dabbous et al. [9] who documented that nearly a quarter of cases (26.7%) were not always compliant to thyroid hormone therapy.

In this study there was a highly significant difference between cases and control groups as regards the parameters of mental (Vinland adaptive and Stanford-Binet5 IQ scores) and physical (height/age, head circumference/age Z scores) development.

Moreover, no significant difference was found between the highly compliant cases and control group as regards the previous parameters. This signifies the importance of adequate compliance to therapy of CH on the neurodevelopmental outcome. And this is in agreement with Babiker et al. [11], Soliman et al. [12], and Azar-Kolakez et al. [13] who concluded that effective screening and adequate treatment and compliance to medication completely assures normal neurodevelopment and linear growth in patients with CH.

Our results have shown statistically high significant positive correlation between compliance score and parameters of physical and mental development. A significant negative correlation was found between the compliance score and the TSH levels at the start of the study and during the follow- up visits. This is in agreement with Grüters and Krude [14], and Kreisner et al. [15] who reported a clinically significant effect of the compliance to therapy on the final physical and IQ outcome in children with CH.


  Conclusion Top


High compliance to treatment of CH was an important prognostic factor for normal mental and physical development in infants and children with CH.

It is recommended to emphasize the importance of early screening for and treatment of CH during the first week of life, to emphasize the importance of compliance of patients and/or their family to therapy of congenital hypothyroidism.

It is recommended to improve the compliance to therapy through health education of both patients and their families and the health-care providers.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Léger J. Congenital hypothyroidism: a clinical update of long-term outcome in young adults. Eur J Endocrinol 2015; 172:R67–R77.  Back to cited text no. 1
    
2.
Rastogi MV, LaFranchi SH. Congenital hypothyroidism. Orphanet J Rare Dis 2010; 5:17.  Back to cited text no. 2
    
3.
Corbetta C, Weber G, Cortinovis F, Calebiro D, Passoni A, Vigone MC, et al. A 7-year experience with low blood TSH cutoff levels for neonatal screening reveals an unsuspected frequency of congenital hypothyroidism. Clin Endocrinol (Oxf) 2009; 71:739–745.  Back to cited text no. 3
    
4.
Bekhit OE, Yousef RM. Permanent and transient congenital hypothyroidism in fayoum, egypt: a descriptive retrospective study. Plos One 2013; 8:e68048.  Back to cited text no. 4
    
5.
Agrawal P, Philip R, Saran S, Gutch M, Razi MS, Agroiya P, et al. Congenital hypothyroidism. Indian J Endocrinol Metab 2015; 19:221–227.  Back to cited text no. 5
    
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Ünüvar T, Demir K, Abacı A, Ataş A, Büyükgebiz A, Böber E. Monitoring and prognostic evaluation of patients with congenital hypothyroidism treated in a pediatric endocrinology unit. Turk J Pediatr 2013; 55:384–390.  Back to cited text no. 6
    
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Dayal D, Prasad R. Congenital hypothyroidism: current perspectives. Res Rep Endocr Disord 2015; 5:91–102.  Back to cited text no. 7
    
8.
Grosse SD, Van Vliet G. Prevention of intellectual disability through screening for congenital hypothyroidism: how much and at what level? Arch Dis Child 2011; 96:374–379.  Back to cited text no. 8
    
9.
Dabbous NI, Abde El-Aziz HM, Abou El– Enein NY, Kandil HH, El-Kafoury AA. Indicators of the screening program for CH in Alexandria. J Egypt Public Health Assoc 2008; 83:307–327.  Back to cited text no. 9
    
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Abdel-Rasoul GM, Hathout HM, Abu Salem ME, El Bahnasy RE, Kasemy ZA. Epidemiological Features of Neonatal hypothyroidism in Menoufiya Governorate, Egypt. (A case–control study). Menoufiya Med J 2011; 24:161–169.  Back to cited text no. 10
    
11.
Babiker AM, Al Jurayyan N, Mohamed SH, Abdullah MA. Overview of diagnosis, management and outcome of congenital hypothyroidism: a call for a national screening programme in Sudan. Sudan J Pediatr 2012; 12:6–17.  Back to cited text no. 11
    
12.
Soliman AT, Azzam S, ElAwwa A, Saleem W, Sabt A. Linear growth and neurodevelopmental outcome of children with congenital hypothyroidism detected by neonatal screening: a controlled study. Indian J Endocrinol Metab 2012; 16:565–568.  Back to cited text no. 12
    
13.
Azar-Kolakez A, Ecosse E, Dos Santos S, Léger J. All-cause and disease-specific mortality and morbidity in patients with congenital hypothyroidism treated since the neonatal period: a national population-based study. J Clin Endocrinol Metab 2013; 98:785–793.  Back to cited text no. 13
    
14.
Gruters A, Krude, H. Detection and treatment of congenital hypothyroidism. Nat Rev Endocrinol 2012; 8:104–113.  Back to cited text no. 14
    
15.
Kreisner MH, Martinez de Mena R, Obregon MJ, Marinkovic D, Howatson A, Visser TJ, et al. Iodothyronine levels in the human developing brain: major regulatory roles of iodothyronine deiodinases in different areas. J Clin Endocrinol Metab 2004; 89:3117–3128.  Back to cited text no. 15
    


    Figures

  [Figure 1]
 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4], [Table 5], [Table 6]



 

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