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Lupu VV, Sasaran MO, Jechel E, Starcea IM, Ioniuc I, Mocanu A, Rosu ST, Munteanu V, Nedelcu AH, Danielescu C, Salaru DL, Knieling A, Lupu A. Celiac disease - a pluripathological model in pediatric practice. Front Immunol 2024; 15:1390755. [PMID: 38715620 PMCID: PMC11074362 DOI: 10.3389/fimmu.2024.1390755] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/23/2024] [Accepted: 04/03/2024] [Indexed: 05/23/2024] Open
Abstract
Being defined as an autoimmune, chronic pathology, frequently encountered in any age group, but especially in pediatrics, celiac disease (also called gluten enteropathy), is gaining more and more ground in terms of diagnosis, but also interest in research. The data from the literature of the last decades attest the chameleonic way of its presentation, there may be both classic onset symptoms and atypical symptoms. Given the impact played by celiac disease, especially in the optimal growth and development of children, the current narrative review aims to highlight the atypical presentation methods, intended to guide the clinician towards the inclusion of the pathology in the differential diagnosis scheme. To these we add the summary presentation of the general data and therapeutic lines regarding the underlying condition and the existing comorbidities. In order to place the related information up to date, we performed a literature review of the recent articles published in international databases. We bring forward the current theories and approaches regarding both classic celiac disease and its atypical manifestations. Among these we note mainly constitutional, skin or mucous, bone, neuro-psychic, renal, reproductive injuries, but also disorders of biological constants and association with multiple autoimmunities. Knowing and correlating them with celiac disease is the key to optimal management of patients, thus reducing the subsequent burden of the disease.
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Affiliation(s)
- Vasile Valeriu Lupu
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Maria Oana Sasaran
- Faculty of Medicine, “George Emil Palade” University of Medicine, Pharmacy, Science and Technology, Targu Mures, Romania
| | - Elena Jechel
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | | | - Ileana Ioniuc
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Adriana Mocanu
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Solange Tamara Rosu
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Valentin Munteanu
- Faculty of Medical Bioengineering, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Alin Horatiu Nedelcu
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Ciprian Danielescu
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Delia Lidia Salaru
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Anton Knieling
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
| | - Ancuta Lupu
- Faculty of Medicine, “Grigore T. Popa” University of Medicine and Pharmacy, Iasi, Romania
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Osteoporosis and Celiac Disease: Updates and Hidden Pitfalls. Nutrients 2023; 15:nu15051089. [PMID: 36904090 PMCID: PMC10005679 DOI: 10.3390/nu15051089] [Citation(s) in RCA: 7] [Impact Index Per Article: 3.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/09/2023] [Revised: 02/15/2023] [Accepted: 02/20/2023] [Indexed: 02/24/2023] Open
Abstract
Celiac disease (CD) is an autoimmune disorder caused by gluten ingestion in genetically predisposed individuals. In addition to the typical gastrointestinal symptoms (e.g., diarrhea, bloating, and chronic abdominal pain), CD may also present with a broad spectrum of manifestations, including low bone mineral density (BMD) and osteoporosis. The etiopathology of bone lesions in CD is multifactorial and other conditions, rather than mineral and vitamin D malabsorption, may affect skeletal health, especially those related to the endocrine system. Here, we describe CD-induced osteoporosis in an attempt to enlighten new and less-known aspects, such as the influence of the intestinal microbiome and sex-related differences on bone health. This review describes the role of CD in the development of skeletal alterations to provide physicians with an updated overview on this debated topic and to improve the management of osteoporosis in CD.
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Aggarwal N, Dwarakanathan V, Singh A, Agarwal A, Khuttan A, Ahmed A, Rajput MS, Chauhan A, Banyal V, Verma AK, Gupta V, Lodha R, Ahuja V, Makharia GK. Spectrum of height in patients with celiac disease. Indian J Gastroenterol 2021; 40:604-612. [PMID: 34921660 DOI: 10.1007/s12664-021-01173-9] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/27/2020] [Accepted: 03/11/2021] [Indexed: 02/04/2023]
Abstract
BACKGROUND Growth retardation is an important feature of celiac disease (CeD) that can lead to the failure of attainment of potential adult height. There is lack of data on the spectrum of height in treatment-naïve patients with CeD, with normal expected height at one end and short stature at the other. METHODS We performed a retrospective analysis of a prospectively maintained database at our center, including a total of 583 treatment-naïve patients with CeD: 419 adults (183 [43.7%] males) and 164 adolescents (12-18 years) (72 [43.9%] males). The details extracted from the database included demographic details, height, weight, body mass index, clinical symptoms, biochemical parameters, anti-tissue transglutaminase antibody anti-tTG Ab) titer, and the severity of villous abnormalities (as per modified Marsh grade). The data from Indian National Family Health Survey-4 were used as comparators. RESULTS Overall, 19.6% of adults and 57.9% of adolescents with CeD had short stature. While mean height of men with CeD was similar, women were taller than population controls. While a higher proportion of men with CeD had short stature as compared to the controls (32.2% vs. 20%, p<0.001), a lower proportion of women with CeD had short stature (9.7% vs. 18.9%, p<0.001). Higher proportion of adolescents with CeD had short stature compared to adults (57.9% vs. 19.6%, p<0.001). On multivariate analysis, adulthood was found to be associated with a lower prevalence of short stature. CONCLUSIONS Overall, 19.6% of adults and 57.9% of adolescents with CeD had short stature. While the mean height of adult men with CeD was not significantly different from the population controls, women were taller. Adolescents with CeD were significantly shorter than their peers.
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Affiliation(s)
- Nishant Aggarwal
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Vignesh Dwarakanathan
- Department of Community Medicine, All India Institute of Medical Sciences, Ansari Nagar, New Delhi 110 029, India
| | - Alka Singh
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Ashish Agarwal
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Akhilesh Khuttan
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
- Department of Internal Medicine, Saint Peter's University Hospital, New Brunswick, NJ, USA
| | - Anam Ahmed
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Mahendra Singh Rajput
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Ashish Chauhan
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Vikas Banyal
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Anil K Verma
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
- Celiac Disease Research Laboratory, Department of Pediatrics, Università Politecnica delle Marche, Ancona, Italy
| | - Vipin Gupta
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
- IBD and Intestinal Failure, Cardiff University Health Board, Cardiff, Wales, UK
| | - Rakesh Lodha
- Department of Paediatrics, All India Institute of Medical Sciences, Ansari Nagar, New Delhi 110 029, India
| | - Vineet Ahuja
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India
| | - Govind K Makharia
- Department of Gastroenterology and Human Nutrition, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, 110 029, India.
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A glimpse into the black box of celiac disease complications: a case report with a rare presentation. GASTROENTEROLOGY REVIEW 2021; 6:111-116. [PMID: 34276837 PMCID: PMC8275971 DOI: 10.5114/pg.2021.106661] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 06/05/2020] [Accepted: 08/10/2020] [Indexed: 11/23/2022]
Abstract
The purpose of this article is to report a case of celiac disease in a child with uncommon presentation and severe complications, and briefly review recent literature regarding celiac disease complications in children. We describe a case report of celiac disease in a child (to our knowledge, this is the first to be reported in its unique presentation in the Russian Federation) and precisely review its presenting complications with the exiting works of literature. Many cases of celiac disease in children who are not diagnosed and treated properly suffer from a plethora of complications due to malabsorption and concurrent autoimmune reactions that affect mainly but are not limited to the endocrine system. Complications of celiac disease in children should always be suspected, and evidence-based follow-up recommendations should be introduced as soon as possible because the burden of celiac disease complications in children is remarkable.
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Sahin Y. Celiac disease in children: A review of the literature. World J Clin Pediatr 2021; 10:53-71. [PMID: 34316439 PMCID: PMC8290992 DOI: 10.5409/wjcp.v10.i4.53] [Citation(s) in RCA: 92] [Impact Index Per Article: 23.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/02/2021] [Revised: 03/23/2021] [Accepted: 05/22/2021] [Indexed: 02/06/2023] Open
Abstract
Celiac disease is an immune-mediated systemic disease triggered by intake of gluten in genetically susceptible individuals. The prevalence of celiac disease in the general population is estimated to be 1% in the world. Its prevalence differs depending on geographical and ethnic variations. The prevalence of celiac disease has increased significantly in the last 30 years due to the increased knowledge and awareness of physicians and the widespread use of highly sensitive and specific diagnostic tests for celiac disease. Despite increased awareness and knowledge about celiac disease, up to 95% of celiac patients still remain undiagnosed. The presentations of celiac disease have significantly changed in the last few decades. Classical symptoms of celiac disease occur in a minority of celiac patients, while older children have either minimal or atypical symptoms. Serologic tests for celiac disease should be done in patients with unexplained chronic or intermittent diarrhea, failure to thrive, weight loss, delayed puberty, short stature, amenorrhea, iron deficiency anemia, nausea, vomiting, chronic abdominal pain, abdominal distension, chronic constipation, recurrent aphthous stomatitis, and abnormal liver enzyme elevation, and in children who belong to specific groups at risk. Early diagnosis of celiac disease is very important to prevent long-term complications. Currently, the only effective treatment is a lifelong gluten-free diet. In this review, we will discuss the epidemiology, clinical findings, diagnostic tests, and treatment of celiac disease in the light of the latest literature.
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Affiliation(s)
- Yasin Sahin
- Pediatric Gastroenterology-Hepatology and Nutrition, Medical Park Gaziantep Hospital, Gaziantep 27560, Turkey
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6
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Growth Hormone Deficiency and Celiac Disease: The Long and Short of It. Indian J Pediatr 2021; 88:536-537. [PMID: 33860884 DOI: 10.1007/s12098-021-03760-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/24/2021] [Accepted: 03/31/2021] [Indexed: 10/21/2022]
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Lapunzina P, Tenorio-Castaño J, Nevado J, Campos Barros Á, Pachajoa H, Ruiz-Pérez VL, Castilla EE. The portrayal of dwarfism without skeletal dysplasia in art: Proportionate short stature due to growth hormone deficiency and other disorders. AMERICAN JOURNAL OF MEDICAL GENETICS PART C-SEMINARS IN MEDICAL GENETICS 2021; 187:186-191. [PMID: 33998134 DOI: 10.1002/ajmg.c.31916] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 03/23/2021] [Revised: 04/08/2021] [Accepted: 04/10/2021] [Indexed: 12/29/2022]
Abstract
In this article, we analyze several works of art which portray individuals with short stature ("dwarfism"). We have focused on eight individuals who we believe have short stature due to growth hormone deficiency (GHD) or closely related disorders, rather than skeletal dysplasia. We discuss them individually, suggest the potential diagnosis, review the characteristics of their life and personal history, and briefly outline the artistic framework in which these works of art were created. This work is a posthumous tribute to the people with short stature portrayed in these works of art, who likely experienced harassment and inappropriate treatment by others and called by derogatory names. We have tried to acknowledge their identities with the respect they deserve.
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Affiliation(s)
- Pablo Lapunzina
- CIBER de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III (ISCIII), Madrid, Spain.,Instituto de Genética Médica y Molecular (INGEMM)-IdiPAZ, Hospital Universitario La Paz, Universidad Autónoma, Madrid, Spain.,ITHACA, European Reference Network on Rare Congenital Malformations and Rare Intellectual Disability, Brussels, Belgium
| | - Jair Tenorio-Castaño
- CIBER de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III (ISCIII), Madrid, Spain.,Instituto de Genética Médica y Molecular (INGEMM)-IdiPAZ, Hospital Universitario La Paz, Universidad Autónoma, Madrid, Spain.,ITHACA, European Reference Network on Rare Congenital Malformations and Rare Intellectual Disability, Brussels, Belgium
| | - Julián Nevado
- CIBER de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III (ISCIII), Madrid, Spain.,Instituto de Genética Médica y Molecular (INGEMM)-IdiPAZ, Hospital Universitario La Paz, Universidad Autónoma, Madrid, Spain.,ITHACA, European Reference Network on Rare Congenital Malformations and Rare Intellectual Disability, Brussels, Belgium
| | - Ángel Campos Barros
- CIBER de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III (ISCIII), Madrid, Spain.,Instituto de Genética Médica y Molecular (INGEMM)-IdiPAZ, Hospital Universitario La Paz, Universidad Autónoma, Madrid, Spain.,ITHACA, European Reference Network on Rare Congenital Malformations and Rare Intellectual Disability, Brussels, Belgium
| | - Harry Pachajoa
- Department of Genetics, Fundación Valle del Lili, Cali, Colombia.,Centro de Investigaciones en Anomalías Congénitas y Enfermedades Raras (CIACER), Universidad ICESI, Cali, Colombia
| | - Víctor L Ruiz-Pérez
- CIBER de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III (ISCIII), Madrid, Spain.,ITHACA, European Reference Network on Rare Congenital Malformations and Rare Intellectual Disability, Brussels, Belgium.,Instituto de Investigaciones Biomédicas "Alberto Sols", Consejo Superior de Investigaciones Científicas (CSIC)-Universidad Autónoma de Madrid (UAM), Madrid, Spain
| | - Eduardo E Castilla
- Latin American Collaborative Study of Congenital Malformations (ECLAMC), FIOCRUZ-Genética, Rio de Janeiro, Brazil.,National Institute of Population Medical Genetics (INAGEMP), Porto Alegre, Brazil
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Setavand Z, Ekramzadeh M, Honar N. Evaluation of malnutrition status and clinical indications in children with celiac disease: a cross-sectional study. BMC Pediatr 2021; 21:147. [PMID: 33781226 PMCID: PMC8006373 DOI: 10.1186/s12887-021-02621-3] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/09/2021] [Accepted: 03/22/2021] [Indexed: 12/19/2022] Open
Abstract
BACKGROUND Celiac Disease (CD) is an autoimmune systemic disorder triggered by gluten in genetically susceptible individuals, which can lead to chronic malabsorption. Considering the changes in the manifestations of CD, this study aimed to determine anthropometric indices and clinical indications in children with CD. METHODS This cross-sectional study aimed to evaluate the children with CD who had referred to Imam Reza Celiac Clinic between 2016 and 2019. Totally, 361 children were eligible and their anti-tissue transglutaminase (TGA-IgA) level, weight, height, and Body Mass Index (BMI) were extracted from their records. The anthropometric indices were presented based on the criteria of the Center for Disease Control and Prevention (CDC) and World Health Organization (WHO). The prevalent symptoms were assessed, as well. RESULTS Based on the CDC's criteria, 18.3, 28.8, and 25.8% of the children had short stature, low body weight, and low BMI, respectively. These measures were obtained as 10, 22.4, and 13.9% according to the WHO's categorization respectively. Furthermore, the most common symptoms among the children were abdominal pain (56.5%), skeletal pain (28%), constipation (27.4%), and anemia (23.8%). CONCLUSION To sum up, the results clearly indicated that growth failure and low height, weight, and BMI were prevalent among the children with CD. Moreover, in addition to gastrointestinal symptoms, a considerable number of patients had skeletal pain and anemia.
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Affiliation(s)
- Zahra Setavand
- Student Research Committee, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Maryam Ekramzadeh
- Nutrition Research Center, Department of Clinical Nutrition, School of Nutrition and Food Sciences, Shiraz University of Medical Sciences, Shiraz, Iran.
| | - Naser Honar
- Neonatal Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
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Prevalence of combined growth hormone deficiency and celiac disease among Saudi Arabian children with short stature: a tertiary care center experience. Chin Med J (Engl) 2020:729-731. [PMID: 32197032 PMCID: PMC7190224 DOI: 10.1097/cm9.0000000000000715] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/01/2022] Open
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10
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Saadah OI, ALNosani NM. Celiac disease in Saudi children with isolated short stature: is it rare or are we not screening rigorously enough? J Pediatr Endocrinol Metab 2020; 33:89-93. [PMID: 31804962 DOI: 10.1515/jpem-2019-0348] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/30/2019] [Accepted: 11/11/2019] [Indexed: 11/15/2022]
Abstract
Background Celiac disease (CeD) is an immune-mediated enteropathy induced by gluten exposure in individuals with genetic susceptibility. Short stature (SS) can be the sole clinical manifestation of CeD, in the absence of gastrointestinal (GI) symptoms. This study aimed to determine the prevalence of CeD in Saudi Arabian children with SS. Patients and methods Medical records were reviewed in a total number of 275 retrospective cases (during the period 2002-2014) of children with isolated SS from King Abdulaziz University Hospital, Jeddah. Their serum samples were tested with tissue transglutaminase (tTG) antibodies. Patients with a positive serology were scheduled for an upper endoscopy and intestinal biopsy to confirm CeD diagnosis before starting a gluten-free diet (GFD). Clinical, anthropometric and laboratory data were recorded for all patients. Results A total of 275 children with SS were included. The mean age ± standard deviation (SD) was 9.4 ± 4.0 years (range, 2.6-16.9 years) and males constituted the predominant gender group (151/275; 54.9%) over females (124/275; 45.1%). The mean ± SD height for age z score (HAZ) was -2.9 ± 1.0.Thirty-eight (13.8%) had positive serology, and 16 (5.8%) had biopsy-proven CeD. Apart from the difference in duration of delayed bone age between CeD patients and CeD-negative serology subjects (mean ± SD, 39.6 ± 10.5 vs. 18.6 ± 16.8, p = 0.02), no other major difference in other clinical or laboratory parameters was evident. Conclusions The prevalence rate of CeD in Saudi Arabian SS children was 5.8%, which is comparable to published reports of a number of other countries. Regular screening of children with SS is therefore justifiable.
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Affiliation(s)
- Omar I Saadah
- Department of Pediatrics, Faculty of Medicine, King Abdulaziz University, Jeddah, Kingdom of Saudi Arabia
| | - Nouf M ALNosani
- Department of Pediatrics, Faculty of Medicine, King Abdulaziz University, Jeddah, Kingdom of Saudi Arabia
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11
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Saadah OI. Short children with impaired growth hormone secretion. Do they have celiac disease? Saudi Med J 2020; 41:68-72. [PMID: 31915797 PMCID: PMC7001070 DOI: 10.15537/smj.2020.1.24785] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/07/2019] [Accepted: 11/24/2019] [Indexed: 11/16/2022] Open
Abstract
OBJECTIVES To determine the prevalence of celiac disease (CeD) in children with short stature (SS) and growth hormone deficiency (GHD). METHODS This is a retrospective study of patients with isolated SS and GHD, diagnosed during the period 2002 to 2016. Their medical records were reviewed and serum tissue transglutaminase (tTG) antibody results retrieved. Patients with positive serology results underwent upper gastrointestinal endoscopy and small bowel biopsy to confirm the diagnosis of CeD. Clinical, anthropometric, and laboratory data were recorded for all patients. RESULTS Of the 351 patients identified with GHD, 199 (56.7%) were male. The mean age±SD was 9.0±3.7 years (range: 2-17.6 years), and the mean±SD height-for-age z score was -2.9±1.3. Partial GHD constituted 42.2% and severe GHD constituted 57.8% of GHD diagnoses. The mean growth hormone (GH) peak level was 5.8±3.9 ng/ml. Forty-seven patients (13.4%) had positive serology, and 14 (4%) had biopsy-proven CeD. No predictors could be identified through binary logistic regression analysis. CONCLUSION A prevalence of CeD seropositivity was found in 13.4% and overt CeD in 4% of children with GHD. The finding of GHD should not preclude the search for CeD, because the majority will potentially improve on a gluten-free diet (GFD).
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Affiliation(s)
- Omar I Saadah
- Pediatric Gastroenterology Unit, Department of Pediatrics, Faculty of Medicine, King Abdulaziz University, Jeddah, Kingdom of Saudi Arabia. E-mail.
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Nardecchia S, Auricchio R, Discepolo V, Troncone R. Extra-Intestinal Manifestations of Coeliac Disease in Children: Clinical Features and Mechanisms. Front Pediatr 2019; 7:56. [PMID: 30891436 PMCID: PMC6413622 DOI: 10.3389/fped.2019.00056] [Citation(s) in RCA: 52] [Impact Index Per Article: 8.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/31/2018] [Accepted: 02/13/2019] [Indexed: 12/11/2022] Open
Abstract
Celiac disease (CD) is a systemic autoimmune disease due to a dysregulated mucosal immune response to gluten and related prolamines in genetically predisposed individuals. It is a common disorder affecting ~1% of the general population, its incidence is steadily increasing. Changes in the clinical presentation have become evident since the 80s with the recognition of extra-intestinal symptoms like short stature, iron deficiency anemia, altered bone metabolism, elevation of liver enzymes, neurological problems. Recent studies have shown that the overall prevalence of extra-intestinal manifestations is similar between pediatric and adult population; however, the prevalence of specific manifestations and rate of improvement differ in the two age groups. For instance, clinical response in children occurs much faster than in adults. Moreover, an early diagnosis is decisive for a better prognosis. The pathogenesis of extra-intestinal manifestations has not been fully elucidated yet. Two main mechanisms have been advanced: the first related to the malabsorption consequent to mucosal damage, the latter associated with a sustained autoimmune response. Importantly, since extra-intestinal manifestations dominate the clinical presentation of over half of patients, a careful case-finding strategy, together with a more liberal use of serological tools, is crucial to improve the detection rate of CD.
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Affiliation(s)
- Silvia Nardecchia
- Department of Medical Translational Sciences and European Laboratory for the Investigation of Food-Induced Diseases, University of Naples Federico II, Naples, Italy
| | - Renata Auricchio
- Department of Medical Translational Sciences and European Laboratory for the Investigation of Food-Induced Diseases, University of Naples Federico II, Naples, Italy
| | | | - Riccardo Troncone
- Department of Medical Translational Sciences and European Laboratory for the Investigation of Food-Induced Diseases, University of Naples Federico II, Naples, Italy
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Walker MD, Zylberberg HM, Green PHR, Katz MS. Endocrine complications of celiac disease: a case report and review of the literature. Endocr Res 2019; 44:27-45. [PMID: 30198791 DOI: 10.1080/07435800.2018.1509868] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
PURPOSE The purpose of this article is to review recent literature regarding endocrine disorders related to celiac disease (CD). METHODS We describe a case report and review existing literature on the endocrine manifestations of CD. RESULTS CD is an autoimmune disorder characterized by intestinal inflammation in response to gluten. CD can cause a wide range of extra-intestinal complications, including endocrine manifestations. Metabolic bone disease including osteoporosis and osteopenia, vitamin D deficiency, secondary hyperparathyroidism and less frequently osteomalacia can be seen. In CD, fracture risk is increased by 30-40%, while risk for hip fracture is approximately doubled. The risk for other endocrine disorders, particularly autoimmune endocrinopathies, is also increased in those with CD compared to the general population. Epidemiologic data indicate the risk for hypothyroidism is 3-4 times higher among those with CD, while risk of type 1 diabetes is greater than double. Risk for primary adrenal insufficiency is a striking 11-fold higher in those with versus without CD, though the absolute risk is low. Fertility is reduced in women with CD before diagnosis by 37% while male fertility in the absence of hypogonadism does not appear to be affected. Other endocrine conditions including hyperthyroidism, ovarian failure, androgen insensitivity, impaired growth and growth hormone deficiency and autoimmune polyendocrine syndromes have also been associated with CD. CONCLUSIONS CD is associated with a wide range of endocrine manifestations.
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Affiliation(s)
- Marcella D Walker
- a Department of Medicine , Columbia University , New York , NY , USA
| | | | - Peter H R Green
- a Department of Medicine , Columbia University , New York , NY , USA
| | - Michael S Katz
- c Department of Medicine , University of Texas Health Science Center at San Antonio , San Antonio , TX , USA
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Araújo IMP, Albuquerque-Souza E, Aguiar-Oliveira MH, Holzhausen M, Oliveira-Neto LA, Salvatori R, Saraiva L, Mayer MPA, Pannuti CM, Ribeiro AO, Romito GA, Pustiglioni FE. Immunological and microbiological periodontal profiles in isolated growth hormone deficiency. J Periodontol 2018; 89:1351-1361. [PMID: 29797719 DOI: 10.1002/jper.17-0687] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/22/2017] [Revised: 04/09/2018] [Accepted: 04/29/2018] [Indexed: 12/15/2022]
Abstract
BACKGROUND Growth hormone (GH) has been identified as an important regulator of the immune response. We have previously shown that adults with isolated GH deficiency (IGHD) due to a mutation in the GH releasing hormone receptor (GHRHR) gene, have a greater chance of having periodontitis. However, the interaction of GH with periodontal tissues is still unknown, and this population has emerged as a unique model to investigate this issue. Therefore, we evaluated the microbiological and immunological periodontal profiles of such individuals. METHODS Nineteen IGHD and 19 controls matched by age, sex, diabetes, and smoking status, were enrolled in this case-control study. Periodontal clinical parameters (probing depth [PD] and clinical attachment loss [AL]) were measured at six sites per tooth. Immune mediators (C-reactive protein, matrix metalloproteinase [MMP]-8, MMP-9, interleukin [IL]-1α, IL-6, IL-8, tumor necrosis factor [TNF]-α, adiponectin, and leptin) were analyzed by enzyme-linked immunosorbent assay (ELISA) in the gingival crevicular fluid (GCF) in four non-adjacent sites for each participant (two with PD ≤3 mm [shallow sites] and two with PD ≥7 mm or the worst PD found in the mouth [deep sites]). Bacterial quantification (Aggregatibacter actinomycetemcomitans, Porphyromonas gingivalis, Treponema denticola, and Tannerella forsythia) of subgingival biofilm samples collected from these same sites was performed by quantitative real-time polymerase chain reaction (qPCR). RESULTS IGHD individuals presented higher values of PD and AL, and increased levels of CRP, IL-8, MMP-8, and adiponectin in the GCF. Bacterial quantification did not identify differences between the two groups. CONCLUSION IGHD alters the local immune response in periodontal pockets leading to greater attachment loss, and GH stands out as an important hormone to be evaluated in the pathogenesis of periodontitis.
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Affiliation(s)
- I M P Araújo
- Division of Periodontics, School of Dentistry, University of São Paulo, SP, Brazil
| | - E Albuquerque-Souza
- Division of Periodontics, School of Dentistry, University of São Paulo, SP, Brazil
| | - M H Aguiar-Oliveira
- Division of Endocrinology, Federal University of Sergipe, 49060-100, Aracaju, SE, Brazil
| | - M Holzhausen
- Division of Periodontics, School of Dentistry, University of São Paulo, SP, Brazil
| | - L A Oliveira-Neto
- Division of Endocrinology, Federal University of Sergipe, 49060-100, Aracaju, SE, Brazil
| | - R Salvatori
- Division of Endocrinology, Department of Medicine, The Johns Hopkins University School of Medicine, Baltimore, MD
| | - L Saraiva
- Division of Periodontics, School of Dentistry, University of São Paulo, SP, Brazil
| | - M P A Mayer
- Department of Microbiology, Institute of Biomedical Sciences, University of São Paulo, SP, Brazil
| | - C M Pannuti
- Division of Periodontics, School of Dentistry, University of São Paulo, SP, Brazil
| | - A O Ribeiro
- Federal University of Sergipe, Division of Immunology and Molecular Biology Laboratory, SE, Brazil
| | - G A Romito
- Division of Periodontics, School of Dentistry, University of São Paulo, SP, Brazil
| | - F E Pustiglioni
- Division of Periodontics, School of Dentistry, University of São Paulo, SP, Brazil
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15
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The pre-treatment characteristics and evaluation of the effects of recombinant human growth hormone therapy in children with growth hormone deficiency and celiac disease or inflammatory bowel disease. Cent Eur J Immunol 2018; 43:69-75. [PMID: 29736148 PMCID: PMC5927175 DOI: 10.5114/ceji.2018.74875] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/08/2017] [Accepted: 12/21/2017] [Indexed: 12/20/2022] Open
Abstract
The aim of the study was to investigate the coincidence of growth hormone deficiency (GHD) and celiac disease (CD) or inflammatory bowel disease (IBD) in patients referred for short stature, and to evaluate the baseline anthropometric parameters and the effectiveness of recombinant human growth hormone (rhGH) therapy in the first year in those patients (GHD+CD/IBD subgroup) in comparison to patients with GHD without CD or IBD (GHD-CD/IBD subgroup). Material and methods The study was retrospective and included 2196 short patients (height SDS [Standard Deviation Score] ≤ –1.2). 1454 patients had height SDS ≤ –2. Twenty-nine patients suffered from CD or IBD. GHD was confirmed in 419 patients with height SDS ≤ –2. The coexistence of GHD and CD or IBD was found in seven patients (GHD+CD/IBD subgroup). Results At baseline the GHD-CD/IBD subgroup did not differ significantly in chronological age, height SDS, height velocity (HV) before rhGH therapy, body weight SDS, and body mass index SDS from the GHD+CD/IBD subgroup. The improvement in height SDS within the first year of rhGH therapy was higher in the GHD+CD/IBD subgroup than in the GHD-CD/IBD subgroup and the difference was statistically significant (p<0.05). HV in the first year of rhGH therapy was also significantly higher in the GHD+CD/IBD subgroup than in the GHD-CD/IBD subgroup (p < 0.05). Conclusions In patients with chronic inflammatory disorders of the gastrointestinal tract, especially celiac disease, coexisting with GHD, rhGH therapy could be effective and should be administered together with therapy of primary gastrointestinal disease.
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16
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Khater D. Endocrinopathies in celiac disease: When the endocrinologist sees what is invisible to the gastroenterologist. ACTA BIO-MEDICA : ATENEI PARMENSIS 2018; 89:117-121. [PMID: 29633735 PMCID: PMC6357610 DOI: 10.23750/abm.v89i1.7119] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Download PDF] [Subscribe] [Scholar Register] [Received: 02/21/2018] [Accepted: 02/21/2018] [Indexed: 12/30/2022]
Abstract
Celiac disease (CD) is a systemic, immune mediated and genetically determined small intestinal disorder characterized by intolerance to dietary gluten that generally presents with gastrointestinal symptoms in young children and extra-intestinal manifestations. Furthermore, there is close association between CD and endocrine diseases, including diabetes, autoimmune thyroid diseases, growth and pubertal disorders, etc. probably due to the presence of a common genetic predisposition. The present review aims to highlight and give more insight to the endocrine changes in CD, especially when there are few or no gastrointestinal symptoms and to emphasize on screening opportunities in some endocrine diseases. (www.actabiomedica.it)
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17
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Freeman HJ. Endocrine manifestations in celiac disease. World J Gastroenterol 2016; 22:8472-8479. [PMID: 27784959 PMCID: PMC5064028 DOI: 10.3748/wjg.v22.i38.8472] [Citation(s) in RCA: 29] [Impact Index Per Article: 3.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/29/2016] [Revised: 08/05/2016] [Accepted: 08/23/2016] [Indexed: 02/06/2023] Open
Abstract
Celiac disease (CD) is an autoimmune small intestinal mucosal disorder that often presents with diarrhea, malabsorption and weight loss. Often, one or more associated endocrine disorders may be associated with CD. For this review, methods involved an extensive review of published English-language materials. In children and adolescents, prospective studies have demonstrated a significant relationship to insulin-dependent or type 1 diabetes, whereas in adults, autoimmune forms of thyroid disease, particularly hypothyroidism, may commonly co-exist. In some with CD, multiple glandular endocrinopathies may also occur and complicate the initial presentation of the intestinal disease. In others presenting with an apparent isolated endocrine disorder, serological screening for underlying subclinical CD may prove to be positive, particularly if type 1 diabetes, autoimmune thyroid or other autoimmune endocrine diseases, such as Addison’s disease are first detected. A number of reports have also recorded hypoparathyroidism or hypopituitarism or ovarian failure in CD and these may be improved with a strict gluten-free diet.
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18
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Snyder J, Butzner JD, DeFelice AR, Fasano A, Guandalini S, Liu E, Newton KP. Evidence-Informed Expert Recommendations for the Management of Celiac Disease in Children. Pediatrics 2016; 138:peds.2015-3147. [PMID: 27565547 DOI: 10.1542/peds.2015-3147] [Citation(s) in RCA: 46] [Impact Index Per Article: 5.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 06/23/2016] [Indexed: 12/11/2022] Open
Abstract
Although the need for effective long-term follow-up for patients with celiac disease (CD) has been recognized by many expert groups, published practice guidelines have not provided a clear approach for the optimal management of these patients. In an attempt to provide a thoughtful and practical approach for managing these patients, a group of experts in pediatric CD performed a critical review of the available literature in 6 categories associated with CD to develop a set of best practices by using evidence-based data and expert opinion. The 6 categories included the following: bone health, hematologic issues, endocrine problems, liver disease, nutritional issues, and testing. Evidence was assessed by using standardized criteria for evaluating the quality of the data, grade of evidence, and strength of conclusions. Over 600 publications were reviewed, and 172 were chosen for inclusion. The thorough review of the results demonstrated that the quality of the data available was often insufficient to provide unequivocal best practices. However, using the available data and the clinical experience of the panel, a practical framework for the management of children with CD was created. These recommendations were developed by our expert panel and do not necessarily reflect the policy of the American Academy of Pediatrics. The potential usefulness of these best practices is underscored by the fact that consensus, measured by the outcome of anonymous voting, was reached by the panel for 24 of the 25 questions. We hope that these best practices may be useful to the pediatric gastroenterology and larger general pediatric communities.
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Affiliation(s)
- John Snyder
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, Children's National Health Systems, Washington, District of Columbia;
| | - J Decker Butzner
- Division of Paediatric Gastroenterology, Hepatology and Nutrition, University of Calgary, Calgary, Alberta, Canada
| | - Amy R DeFelice
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, New York-Presbyterian Hospital, Columbia University, New York, New York
| | - Alessio Fasano
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, Massachusetts General Hospital for Children, Boston, Massachusetts
| | - Stefano Guandalini
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, University of Chicago Medical Center, Chicago, Illinois
| | - Edwin Liu
- Digestive Health Institute, Children's Hospital Colorado, Aurora, Colorado; and
| | - Kimberly P Newton
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, Rady Children's Hospital and University of California San Diego School of Medicine, San Diego, California
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Hawkes CP, Grimberg A. Insulin-Like Growth Factor-I is a Marker for the Nutritional State. PEDIATRIC ENDOCRINOLOGY REVIEWS : PER 2015; 13:499-511. [PMID: 26841638 PMCID: PMC5576178] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Subscribe] [Scholar Register] [Indexed: 06/05/2023]
Abstract
Measurement of the serum concentration of insulin-like growth factor-I (IGF-l) is generally used as a screening investigation for disorders of the growth hormone (GH)/IGF-I axis in children and adolescents with short stature. IGF-I concentration is sensitive to short-term and chronic alterations in the nutritional state, and the interpretation of IGF-I measurements requires knowledge of the child's nutritional status. In this review, we summarize the effects of nutrition on the GH/IGF-I axis, and review the clinical implications of these interactions throughout childhood, both in under-nutrition and over-nutrition.
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Affiliation(s)
- Colin P Hawkes
- Division of Endocrinology and Diabetes, The Children’s Hospital of Philadelphia, Philadelphia, PA, USA
- Department of Paediatrics and Child Health, University College Cork, Cork, Ireland
- The National Children’s Research Centre, Dublin, Ireland
| | - Adda Grimberg
- Division of Endocrinology and Diabetes, The Children’s Hospital of Philadelphia, Philadelphia, PA, USA
- Department of Pediatrics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA 19104, USA
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20
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Bozzola M, Meazza C, Villani A. Auxo-Endocrinological Approach to Celiac Children. Diseases 2015; 3:111-121. [PMID: 28943613 PMCID: PMC5548236 DOI: 10.3390/diseases3020111] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/23/2015] [Revised: 05/29/2015] [Accepted: 06/01/2015] [Indexed: 11/24/2022] Open
Abstract
Celiac disease is a permanent genetically determined intolerance to gluten that generally presents with gastrointestinal symptoms in young children and extraintestinal manifestations (endocrinological, dermatological, neurological, etc.) later. Furthermore, many studies demonstrate the close association between celiac and endocrine diseases, including growth and pubertal disorders, type I diabetes mellitus and autoimmune thyroid diseases, probably due to the presence of a common genetic predisposition. Follow-up for celiac children after the start of gluten-free diet is mandatory to avoid complications such as growth hormone deficiency. The present review deals with the problem of the diagnosis of endocrine-associated diseases in celiac children and gives suggestions for correct management and follow-up of these patients.
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Affiliation(s)
- Mauro Bozzola
- Internal Medicine and Therapeutics Department, Pediatrics and Adolescentology Unit, University of Pavia, Fondazione IRCCS San Matteo, Viale Golgi 19, 27100 Pavia, Italy.
| | - Cristina Meazza
- Internal Medicine and Therapeutics Department, Pediatrics and Adolescentology Unit, University of Pavia, Fondazione IRCCS San Matteo, Viale Golgi 19, 27100 Pavia, Italy.
| | - Alberto Villani
- Department of Pediatrics, Pediatric and Infectious Diseases Unit, Bambino Gesù Children Hospital, IRCCS, Piazza Sant'Onofrio 4, 00165 Rome, Italy.
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21
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Naderi F, Eslami SR, Mirak SA, Khak M, Amiri J, Beyrami B, Shekarchi B, Poureisa M. Effect of growth hormone deficiency on brain MRI findings among children with growth restrictions. J Pediatr Endocrinol Metab 2015; 28:117-23. [PMID: 25153566 DOI: 10.1515/jpem-2013-0294] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/13/2013] [Accepted: 07/14/2014] [Indexed: 01/26/2023]
Abstract
OBJECTIVES Growth hormone deficiency (GHD) is a major problem among children with short stature. In this study, the role of brain magnetic resonance imaging (MRI) in defining the underlying defects among short children with GHD is evaluated. METHODS In a cross-sectional study, data of 158 children were evaluated. Growth hormone (GH) levels were measured using stimulating tests and brain MRI with gadolinium contrast was applied, as well. RESULTS Some 25.3% of patients had GHD with a mean age of 8.01±3.40 years. MRI results showed 35 as normal, four with pituitary hypoplasia, and one with microadenoma. The MRI results were significantly associated with GH levels and presence of other endocrine disorders. There was a significant association between prenatal disorders and patients' bone age delay. CONCLUSIONS In patients with severe GHD and patients with multiple pituitary hormone deficiencies, MRI is more likely to be abnormal, and bone age is much delayed in patients with history of prenatal disorders.
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22
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Meazza C, Pagani S, Gertosio C, Bozzola E, Bozzola M. Celiac disease and short stature in children. Expert Rev Endocrinol Metab 2014; 9:535-542. [PMID: 30736215 DOI: 10.1586/17446651.2014.932248] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/08/2022]
Abstract
Celiac disease (CD) is a genetically determined gluten-sensitive enteropathy resulting in nutrient malabsorption, with an increasing incidence worldwide. In CD children, short stature may be the only presenting clinical feature, even in the absence of gastrointestinal symptoms. Generally, a gluten-free diet (GFD) leads to rapid catch-up growth within 1-2 years. The pathogenesis of CD-associated short stature is still unclear. Besides the involvement of the growth hormone (GH)/IGF-I axis, other pathogenetic mechanisms may include autoimmune disorders of the pituitary gland and altered ghrelin secretion. Furthermore, some CD patients do not show catch-up growth during a GFD, despite reversion to seronegativity for CD markers. These subjects may have GH deficiency and could benefit from GH therapy. This review deals with the problem of linear growth in CD children and points to the importance of the evaluation of GH secretion in those children who show no catch-up growth after the introduction of a GFD.
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Affiliation(s)
- Cristina Meazza
- a Internal Medicine and Therapeutics Department, Pediatrics and Adolescentology Unit, University of Pavia, Fondazione IRCCS Policlinico San Matteo, Viale Golgi 19, 27100 Pavia, Italy
| | - Sara Pagani
- a Internal Medicine and Therapeutics Department, Pediatrics and Adolescentology Unit, University of Pavia, Fondazione IRCCS Policlinico San Matteo, Viale Golgi 19, 27100 Pavia, Italy
| | - Chiara Gertosio
- b Fondazione IRCCS Policlinico San Matteo, Viale Golgi 19, 27100 Pavia, Italy
| | - Elena Bozzola
- c Department of Pediatrics, Pediatric and Infectious Diseases Unit, Bambino Gesù Children's Hospital, IRCCS, Piazza Sant' Onofrio 4, 00165 Rome, Italy
| | - Mauro Bozzola
- a Internal Medicine and Therapeutics Department, Pediatrics and Adolescentology Unit, University of Pavia, Fondazione IRCCS Policlinico San Matteo, Viale Golgi 19, 27100 Pavia, Italy
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23
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Krupa-Kozak U. Pathologic bone alterations in celiac disease: etiology, epidemiology, and treatment. Nutrition 2014; 30:16-24. [PMID: 24290593 DOI: 10.1016/j.nut.2013.05.027] [Citation(s) in RCA: 52] [Impact Index Per Article: 4.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/15/2013] [Revised: 05/29/2013] [Accepted: 05/31/2013] [Indexed: 12/13/2022]
Abstract
Low bone mineral density (BMD), osteopenia, and osteoporosis are frequent complications of celiac disease (CD). The etiology of pathologic bone alterations in CD is multifactorial; however, two main mechanisms are involved: intestinal malabsorption and chronic inflammation. A strict gluten-free diet (GFD) is thought to be the only effective treatment for CD; but treating bone complications related to CD remains complex. The objective of this review is to elucidate the bones problems related to CD and to increase awareness of osteoporosis development, considered as a sign of atypical CD presentation. Currently, a question of whether GFD alone is an effective treatment to correct the bone alterations in patients with CD is under debate. This review presents factors contributing to pathologic bone derangement, recent research on the epidemiology of low BMD, osteoporosis, and fractures, and the treatment of bone problems in patients with CD. The roles of calcium and transport mechanisms are additionally presented.
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Affiliation(s)
- Urszula Krupa-Kozak
- Institute of Animal Reproduction and Food Research of the Polish Academy of Sciences, Department of Chemistry and Biodynamics of Food, Olsztyn, Poland.
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24
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Delvecchio M, Faienza MF, Lonero A, Rutigliano V, Francavilla R, Cavallo L. Prolactin may be increased in newly diagnosed celiac children and adolescents and decreases after 6 months of gluten-free diet. Horm Res Paediatr 2014; 81:309-313. [PMID: 24603159 DOI: 10.1159/000357064] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/15/2013] [Accepted: 10/31/2013] [Indexed: 12/17/2022] Open
Abstract
BACKGROUND/AIMS Prolactin (PRL) is produced by the anterior pituitary gland. It exerts its role on the breast gland but also plays a modulatory role in autoimmune mechanisms. Celiac disease (CD) is a gluten-sensitive autoimmune enteropathy sometimes associated with autoimmune endocrinopathies. No data on PRL levels in CD patients are available at diagnosis, and no conclusive data are reported. METHODS We aimed to evaluate PRL secretion in newly diagnosed CD pediatric patients and, in the case of hyperprolactinemia, any changes in its levels while the patients were on a gluten-free diet (GFD). We recruited 67 patients and 39 healthy controls. RESULTS PRL was statistically higher in the CD patients (13.5±9.2 ng/ml) than in the controls (8.5±5.0 ng/ml). In the CD group, PRL was inversely correlated with the age at diagnosis (r=-0.326; p=0.007). In patients with hyperprolactinemia at diagnosis, PRL decreased after 6 months of GFD. CONCLUSION This paper confirms that PRL may be increased at diagnosis of CD and shows, for the first time, that it decreases after a short course of GFD. Changes in the levels of inflammatory cytokines in CD may account for changes in PRL levels. Younger patients seem more prone to develop hyperprolactinemia than older ones.
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Affiliation(s)
- Maurizio Delvecchio
- Pediatrics Unit, IRCCS Casa Sollievo della Sofferenza, San Giovanni Rotondo, Italy
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25
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Bergfeld WF. Growth hormone deficiency in a young patient with alopecia areata. J Investig Dermatol Symp Proc 2013; 16:S54-S55. [PMID: 24326559 DOI: 10.1038/jidsymp.2013.21] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/03/2023]
Abstract
A young female child aged 7 years presented initially with chronic alopecia areata, which over 2 years progressed to alopecia areata universalis.
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Affiliation(s)
- Wilma F Bergfeld
- Department of Dermatology, Cleveland Clinic, Cleveland, Ohio, USA
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26
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Sisley S, Trujillo MV, Khoury J, Backeljauw P. Low incidence of pathology detection and high cost of screening in the evaluation of asymptomatic short children. J Pediatr 2013; 163:1045-51. [PMID: 23706358 PMCID: PMC3786014 DOI: 10.1016/j.jpeds.2013.04.002] [Citation(s) in RCA: 64] [Impact Index Per Article: 5.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/03/2012] [Revised: 03/14/2013] [Accepted: 04/04/2013] [Indexed: 10/26/2022]
Abstract
OBJECTIVE To determine the incidence of pathology during routine screening of healthy short children, testing adherence to a consensus statement on the diagnosis and treatment of children with idiopathic short stature, and the cost per identified diagnosis resulting from comprehensive screening. STUDY DESIGN Retrospective chart review of 1373 consecutive short stature referrals evaluated at the Cincinnati Children's Hospital Medical Center Pediatric Endocrinology Clinic between 2008 and 2011. We identified 235 patients with a height of <3rd percentile, negative history and review of systems, and normal physical examination. Outcome measures were incidence of pathology detection, diagnostic group characteristics, clinicians' adherence to testing guidelines, and screening costs. ANOVA and χ(2) were used to analyze the data. RESULTS Nearly 99% of patients were diagnosed as possible variants of normal growth: 23% with familial short stature, 41% with constitutional delay of growth and maturation, and 36% with idiopathic short stature. The incidence of newly diagnosed pathology was 1.3%: 1 patient with biopsy-proved celiac disease, 1 with unconfirmed celiac disease, and 1 with potential insulin-like growth factor I receptor defect. On average, each patient had 64.3% of the recommended tests for age and sex; 2.1% of patients had all of the recommended testing. The total screening tests costs were $315321, yielding $105107 per new diagnosis entertained. CONCLUSIONS Healthy short children do not warrant nondirected, comprehensive screening. Future guidelines for evaluating short stature should include patient-specific testing.
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Affiliation(s)
- Stephanie Sisley
- Division of Pediatric Endocrinology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH.
| | | | - Jane Khoury
- Divisions of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH
| | - Philippe Backeljauw
- Divisions of Pediatric Endocrinology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH
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27
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Bone mineralization in celiac disease. Gastroenterol Res Pract 2012; 2012:198025. [PMID: 22737164 PMCID: PMC3378976 DOI: 10.1155/2012/198025] [Citation(s) in RCA: 42] [Impact Index Per Article: 3.2] [Reference Citation Analysis] [Abstract] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/06/2012] [Revised: 03/12/2012] [Accepted: 04/03/2012] [Indexed: 01/31/2023] Open
Abstract
Evidence indicates a well-established relationship between low bone mineral density (BMD) and celiac disease (CD), but data on the pathogenesis of bone derangement in this setting are still inconclusive. In patients with symptomatic CD, low BMD appears to be directly related to the intestinal malabsorption. Adherence to a strict gluten-free diet (GFD) will reverse the histological changes in the intestine and also the biochemical evidence of calcium malabsorption, resulting in rapid increase of BMD. Nevertheless, GFD improves BMD but does not normalize it in all patients, even after the recovery of intestinal mucosa. Other mechanisms of bone injury than calcium and vitamin D malabsorption are thought to be involved, such as proinflammatory cytokines, parathyroid function abnormalities, and misbalanced bone remodeling factors, most of all represented by the receptor activator of nuclear factor B/receptor activator of nuclear factor B-ligand/osteoprotegerin system. By means of dual-energy X-ray absorptiometry (DXA), it is now rapid and easy to obtain semiquantitative values of BMD. However, the question is still open about who and when submit to DXA evaluation in CD, in order to estimate risk of fractures. Furthermore, additional information on the role of nutritional supplements and alternative therapies is needed.
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COSTACURTA M, CONDÒ R, SICURO L, PERUGIA C, DOCIMO R. Cervical vertebral maturation and dental age in coeliac patients. ORAL & IMPLANTOLOGY 2011; 4:11-17. [PMID: 23277868 PMCID: PMC3530968] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Subscribe] [Scholar Register] [Indexed: 06/01/2023]
Abstract
AIM The aim of the study was to evaluate the cervical vertebral maturation and dental age, in group of patients with coelic disease (CD), in comparison with a control group of healthy subjects. METHODS At the Paediatric Dentistry Unit of PTV Hospital, "Tor Vergata" University of Rome, 120 female patients, age range 12.0-12.9 years were recruited. Among them, 60 subjects (Group 1) were affected by CD, while the control group (Group 2) consisted of 60 healthy subjects, sex and age-matched. The Group 1 was subdivided, according to the period of CD diagnosis, in Group A (early diagnosis) and Group B (late diagnosis). The skeletal age was determined by assessing the cervical vertebral maturation, while the dental age has been determined using the method codified by Demirjiyan. STATISTICS.: The analyses were performed using the SPSS software (version 16; SPSS Inc., Chicago IL, USA). In all the assessments a significant level of alpha = 0.05 was considered. RESULTS There are no statistically significant differences between Group 1 and Group 2 as for chronological age (p=0.122). Instead, from the assessment of skeletal-dental age, there are statistically significant differences between Group 1 - Group 2 (p<0.001) and Group A - Group B (p<0.001). The statistical analysis carried out to assess the differences between chronological and skeletal-dental age within the single groups, show a statistically significant difference in Group 1 (p<0.001) and in Group B (p<0.001), while there are no statistically significant differences in Group 2 (p=0.538) and in Group A (p=0.475). A correlation between skeletal and dental age was registered; for Groups 1-2 and for Groups A-B the Pearson coefficient was respectively equal to 0.967 and 0.969, with p<0.001. Through the analysis of data it is possible to assess that the percentage of subjects with skeletal and dental age delay corresponds to 20% in healthy subjects, 56.7% in coeliac subjects, 23% in coeliac subjects with early diagnosis and 90% in coeliac subjects with late diagnosis. From the comparison between Group 2 and Group A there are no statistically significant differences (p=0.951). Conclusions. The skeletal age and dental age delay may be two predictive indexes for a CD diagnosis. The dental age and cervical vertebral maturity can be assessed with a low cost, non invasive, easy to perform exam carried out through the routine radiographic examinations such as orthopanoramic and lateral teleradiography.
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Affiliation(s)
- M. COSTACURTA
- Department of Dentistry, Paediatric Dentistry Unit, University of Rome “Tor Vergata”, Rome, Italy
| | - R. CONDÒ
- Department of Dentistry, Paediatric Dentistry Unit, University of Rome “Tor Vergata”, Rome, Italy
| | - L. SICURO
- Researcher of the Italian National Institute of Statistics (ISTAT)
| | - C. PERUGIA
- Department of Dentistry, Paediatric Dentistry Unit, University of Rome “Tor Vergata”, Rome, Italy
| | - R. DOCIMO
- Department of Dentistry, Paediatric Dentistry Unit, University of Rome “Tor Vergata”, Rome, Italy
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Clinical utility of serologic testing for celiac disease in asymptomatic patients: an evidence-based analysis. ONTARIO HEALTH TECHNOLOGY ASSESSMENT SERIES 2011; 11:1-63. [PMID: 23074415 PMCID: PMC3377551] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Subscribe] [Scholar Register] [Indexed: 06/01/2023]
Abstract
OBJECTIVE The objective of this evidence-based analysis was to evaluate the clinical utility of serologic testing for celiac disease in asymptomatic individuals presenting with one of the non-gastrointestinal conditions evaluated in this report. The clinical utility was based on the effects of a gluten-free diet (GFD) on outcomes specific to each of these conditions. The prevalence of celiac disease in asymptomatic individuals and one of these non-gastrointestinal conditions was also evaluated. CLINICAL NEED AND TARGET POPULATION CELIAC DISEASE: Celiac disease is an autoimmune disease characterized by a chronic inflammatory state of the proximal small bowel mucosa accompanied by structural and functional changes. TECHNOLOGY UNDER EVALUATION: SEROLOGIC TESTS FOR CELIAC DISEASE: There are a number of serologic tests for celiac disease available. Serologic tests are automated with the exception of the anti-endomysial antibody test, which is more time-consuming and operator-dependent than the other tests. RESEARCH QUESTIONS What is the prevalence of asymptomatic celiac disease in patients presenting with one of the non-gastrointestinal conditions evaluated?What is the effect of the gluten-free diet on condition-specific outcomes in patients with asymptomatic celiac disease presenting with one of the non-gastrointestinal conditions evaluated?What is the clinical utility of serologic testing for celiac disease in asymptomatic patients presenting with one of the non-gastrointestinal conditions evaluated? The clinical utility was defined as the impact of the GFD on disease specific outcomes.What is the risk of all-cause mortality and lymphoma in individuals with asymptomatic celiac disease?What is the budget impact of serologic testing for celiac disease in asymptomatic subjects presenting with one of the non-gastrointestinal conditions evaluated? RESEARCH METHODS STUDY POPULATION The study population consisted of individuals with newly diagnosed celiac disease without any symptoms consistent with the disease presenting with one of the non-gastrointestinal conditions evaluated. When evaluating the risk of lymphoma and all-cause mortality, the study population consisted of asymptomatic individuals with a positive celiac disease serologic test and/or small bowel biopsy. LITERATURE SEARCH SEARCH STRATEGY Literature searches were performed for each disease/condition evaluated between December 2010 and March 2011 using OVID MEDLINE, the Cochrane Library, and the International Agency for Health Technology Assessment (INAHTA). No restrictions for start date of search were used. Abstracts were reviewed by a single reviewer and, for those studies meeting the eligibility criteria, full-text articles were obtained. Reference lists were also examined for any additional relevant studies not identified through the search. Articles with an unknown eligibility were reviewed with a second clinical epidemiologist and then a group of epidemiologists until consensus was established. INCLUSION CRITERIA Studies, systematic reviews, and meta-analyses that assessed the effects of a GFD in patients with newly diagnosed asymptomatic celiac disease presenting with one of the non-gastrointestinal conditions evaluated. If symptoms were not reported in the study but subjects were identified through screening for celiac disease the study was included.Studies, systematic reviews, and meta-analyses that assessed the prevalence of newly diagnosed asymptomatic celiac disease in patients with one of the non-gastrointestinal conditions evaluated. If symptoms were not reported in the study but subjects were identified through screening for celiac disease the study was included.Studies, systematic reviews, and meta-analyses that evaluated the risk of all-cause mortality or lymphoma in individuals with asymptomatic celiac disease.Sample size ≥ 10.Publications in English. EXCLUSION CRITERIA Studies that retrospectively assessed the prevalence of asymptomatic celiac disease.Studies that reported the prevalence of one of the non-gastrointestinal conditions evaluated in subjects already diagnosed with celiac disease.Studies in individuals with one of the non-gastrointestinal conditions evaluated if the condition could be explained by other causes.Studies in subjects with celiac disease and symptoms consistent with the disease. If the study included individuals with and without symptoms consistent with celiac disease and their results were analysed separately, the results in individuals without symptoms were included in the analysis.Studies in which individuals did not report any symptoms consistent with celiac disease at study start but that either retrospectively reported the presence of such symptoms after following a GFD, or that previously presented with symptoms consistent with celiac disease.Study results published in letters to the editor or comments about other studies.Studies with a sample size ≥ 10, however, in which less than 10 patients were included in the analysis. OUTCOMES OF INTEREST The effects of a GFD on disease-specific outcomes for each condition evaluated in patients with asymptomatic celiac disease was assessed. The prevalence of asymptomatic celiac disease in patients presenting with one of the conditions evaluated was also assessed. RESULTS OF EVIDENCE-BASED ANALYSIS: Three eligible observational studies evaluated the effects of GFD on growth parameters in subjects with asymptomatic celiac disease and idiopathic short stature. Four eligible observational studies evaluated the effects of GFD on metabolic control in subjects with asymptomatic celiac disease and type 1 diabetes. Five eligible observational studies evaluated the risk of all-cause mortality and five eligible observational studies evaluated the risk of lymphoma in subjects with asymptomatic celiac disease. No eligible studies on the effects of the GFD for the other conditions evaluated were identified. Twenty-three eligible studies measured the prevalence of asymptomatic celiac disease in subjects presenting with one of the conditions evaluated. PREVALENCE OF CELIAC DISEASE IN ASYMPTOMATIC PATIENTS: The prevalence of celiac disease in asymptomatic patients presenting with one of the conditions evaluated was analysed. Most studies also included a control group that generally consisted of individuals randomly selected from the general population. Although there was a trend to a higher prevalence of asymptomatic celiac disease in individuals with the conditions evaluated compared to the controls, it only reached statistical significance in type 1 diabetes. No eligible prevalence studies were identified in patients with amenorrhea, delayed puberty, alopecia, and depression. THE EFFECTS OF A GLUTEN-FREE DIET ON DISEASE-SPECIFIC OUTCOMES IN PATIENTS WITH ASYMPTOMATIC CELIAC DISEASE: THE EFFECTS OF GFD ON METABOLIC CONTROL IN PATIENTS WITH ASYMPTOMATIC CELIAC DISEASE AND TYPE 1 DIABETES: The effects of a GFD on metabolic control (HbA1c, number of hypoglycemic episodes, and changes in insulin dosage) in subjects with asymptomatic celiac disease and type 1 diabetes were evaluated. One prospective case-control study reported an increase in HbA1c levels in cases with type 1 diabetes and asymptomatic celiac disease after the introduction of a GFD, however, the clinical significance of this change is unclear. Only one eligible retrospective case-control study evaluated the effects of a GFD on hypoglycemia episodes and since there were inadequate details in the study about both the ascertainment and severity of hypoglycemia episodes in both cases and controls, it is not possible to draw conclusions regarding the effects of a GFD on hypoglycemia episodes based on this study. One prospective case-control study did not show a statistically significant change in insulin dosage between cases with type 1 diabetes and asymptomatic celiac disease and controls with type 1 diabetes either before or after the introduction of a GFD. No eligible studies that evaluated the effects of a GFD on the long-term outcomes of type 1 diabetes such as cardiovascular or renal events in patients with asymptomatic celiac disease were identified. THE EFFECTS OF A GLUTEN-FREE DIET IN PATIENTS WITH IDIOPATHIC SHORT STATURE AND ASYMPTOMATIC CELIAC DISEASE: A total of 3 eligible studies were identified. All studies consisted of case series that compared growth parameters in subjects with asymptomatic celiac disease and idiopathic short stature before and after the celiac disease was diagnosed and the GFD was instituted. Most subjects included in the studies demonstrated an improvement in growth parameters. Compliance with the GFD was not reported in the studies. The results of the studies suggest an increase in growth velocity in pediatric patients with asymptomatic celiac disease and idiopathic short stature once a GFD is introduced. RISK OF LYMPHOMA IN PATIENTS WITH ASYMPTOMATIC CELIAC DISEASE: One retrospective cohort study evaluated the risk of lymphoma in patients with asymptomatic celiac disease. The authors concluded that the number of events identified was low during the long follow-up period and that the risk of overall malignancies was not increased among patients with asymptomatic celiac disease. RISK OF ASYMPTOMATIC CELIAC DISEASE IN PATIENTS WITH LYMPHOMA: Four case-control studies, one of which retrospective, evaluated the risk of asymptomatic celiac disease in patients newly diagnosed with lymphoma. One retrospective cohort study did not show an increase in the risk of lymphoma among subjects with asymptomatic celiac disease. Three prospective case-control studies did not find a statistically significant risk of asymptomatic celiac disease in patients with newly diagnosed lymphoma. (ABSTRACT TRUNCATED)
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Rana KS, Puri P, Badwal S. Prevalence of Celiac Disease in Children with Unexplained Failure to Thrive. Med J Armed Forces India 2010; 66:134-7. [PMID: 27365725 PMCID: PMC4920909 DOI: 10.1016/s0377-1237(10)80125-x] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/29/2008] [Accepted: 02/26/2010] [Indexed: 11/17/2022] Open
Abstract
BACKGROUND Gluten sensitive enteropathy or celiac disease (CD) is a disorder of small bowel that occurs upon exposure to gluten. A total of 67 children of either sex in the age group of 1-12 years with unexplained failure to thrive were studied for the prevalence of CD. METHODS This was a cross-sectional study. It included detailed history, clinical assessment, estimation of anti gliadin (AGA), tissue transglutaminase antibodies (tTGA) and duodenal biopsy. Treatment with gluten free diet and follow-up of diagnosed cases was done for one year. RESULT Sixteen cases (23.88%) had villous atrophy and positive serology, essential criteria for the diagnosis of CD. Forty six (69%) children were between 4-12 years of age. Male to female ratio was 2.3:1. Main symptoms were irritability (63%), diarrhea (56%) and weight loss (56%). Thirty seven (56%) children had weight less than 3(rd) percentile. tTGA was 100% sensitive and 90.2% specific. Duodenal biopsy showed decreased villious-crypt ratio in 81.25% and intra epithelial lymphocytosis in 81% children (p<0.000001). All the confirmed cases were advised strict gluten free diet for one year. On follow-up at six months, all children showed improvement in their symptoms and weight gain. CONCLUSION CD is an important cause of unexplained failure to thrive in children.
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Affiliation(s)
- KS Rana
- Senior Advisor (Paediatrics and Paediatric Neurology), Army Hospital (R&R), Delhi Cant, New Delhi-10
| | - P Puri
- Senior Advisor (Medicine and Gastroenterology), Army Hospital (R&R), Delhi Cant, New Delhi-10
| | - S Badwal
- Classified Specialist (Pathology and Nephropathology), 151 Base Hospital, C/o 99 APO
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Delvecchio M, De Bellis A, Francavilla R, Rutigliano V, Predieri B, Indrio F, De Venuto D, Sinisi AA, Bizzarro A, Bellastella A, Iughetti L, Cavallo L. Anti-pituitary antibodies in children with newly diagnosed celiac disease: a novel finding contributing to linear-growth impairment. Am J Gastroenterol 2010; 105:691-696. [PMID: 19904244 DOI: 10.1038/ajg.2009.642] [Citation(s) in RCA: 34] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
OBJECTIVES The possible autoimmune involvement of the pituitary gland in patients with celiac disease (CD) has been suggested but demonstrated in only a few patients on gluten-free diet. We aimed to assess the prevalence and clinical meaning of anti-pituitary antibodies (APA) in children and adolescents with the newly diagnosed CD. METHODS A total of 119 patients with CD (0.9-15.8 years old) attending the inpatient clinic of University Hospital were recruited for the cross-sectional study. Their height, weight, and body mass index (BMI) were recorded, and insulin-like growth factor-1 (IGF-1) and APA were assayed. APA was also determined in 98 sex- and age-matched controls. RESULTS APA were detected in 50 patients (42.0%), 15 of them with high titer (30%) and 35 with low titer (70%), and in 2 control subjects at low titer (2%) (P<0.001). IGF-1 was higher in patients with negative than with low titer (P=0.02) or high titer APA (P=0.03). Height was more reduced in high-titer APA patients than in the negative ones (P<0.01). Height was positively correlated with IGF-1 (P<0.01) and negatively with chronological age (P=0.001). IGF-1 was positively correlated with BMI (P<0.001). For height prediction the regression analysis showed the rank order 1 for chronological age and 2 for IGF-1. CONCLUSIONS In this paper we have shown a remarkable prevalence of positive APA in newly diagnosed CD patients. High APA titers are associated with height impairment, likely mediated by a reduction of IGF-1, thus suggesting that autoimmune pituitary process could induce a linear-growth impairment.
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Affiliation(s)
- Maurizio Delvecchio
- Unità Operativa Complessa di Pediatria, IRCCS Casa Sollievo della Sofferenza, San Giovanni Rotondo, Italy.
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Nemet D, Raz A, Zifman E, Morag H, Eliakim A. Short stature, celiac disease and growth hormone deficiency. J Pediatr Endocrinol Metab 2009; 22:979-83. [PMID: 20020588 DOI: 10.1515/jpem.2009.22.10.979] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/20/2022]
Abstract
Celiac disease (CD) is a prevalent, genetically determined, autoimmune, chronic inflammatory state caused by intolerance to gluten that results mainly in gastrointestinal manifestations. One of the most common extra-intestinal manifestations of CD is short stature, and in some patients, short stature may be the presenting and only symptom of the disease, making the diagnosis of CD challenging. Impaired growth in children with CD results mainly from nutritional deficits, and withdrawal of gluten from the diet is frequently associated with a marked improvement of linear growth. In some patients, CD may be characterized by growth hormone (GH) resistance, as suggested by normal or elevated GH levels and low insulin-like growth factor-I (IGF-I) levels. Rarely, it has been shown that poor catch-up growth and/or IGF-I response to gluten-free diet may be due to the coexistence of celiac disease and GH deficiency. We present two children with coexisting CD and GH deficiency. One patient had MRI findings suggesting congenital isolated GH deficiency, and a possibility of developing multiple pituitary hormone deficiencies later in life.
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Affiliation(s)
- D Nemet
- Gastrointestinal and Endocrine Clinic, Pediatric Department, Meir Medical Center, Kfar Saba, Israel
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Gueiros ACLN, Silva GAP. Soropositividade para doença celíaca em crianças e adolescentes com baixa estatura. REVISTA PAULISTA DE PEDIATRIA 2009. [DOI: 10.1590/s0103-05822009000100005] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/26/2023]
Abstract
OBJETIVO: Avaliar a frequência da positividade do marcador sorológico para doença celíaca em crianças e adolescentes com baixa estatura, utilizando-se o anticorpo anti-transglutaminase humana como teste de triagem. MÉTODOS: Estudo descritivo com amostra obtida por conveniência. Foi realizado no período de abril a setembro de 2004 no Ambulatório Geral de Pediatria do Instituto Materno Infantil Professor Fernando Figueira e no Ambulatório de Crescimento e Desenvolvimento do Hospital das Clínicas. Foram considerados casos as crianças e os adolescentes portadores de baixa estatura, definida como aquela abaixo do percentil 3 para idade e sexo, utilizando como referência o gráfico de altura/idade do National Center for Health Statistics, 2000. Foi pesquisado o anticorpo anti-transglutaminase humana (AATGh), considerado positivo se concentração >20U/mL e, nos positivos, o anticorpo antiendomísio (AAE). RESULTADOS: Foram avaliados 78 pacientes, sendo 41 (53%) do sexo feminino. O AATGh foi positivo em 3/78 (3,8%) dos pacientes. O AAE foi positivo em um paciente, naquele com concentração mais elevada do AATGh. Considerando-se a positividade para os dois testes, a soropositividade foi de 1,3%. CONCLUSÕES: A presença de marcador sorológico para doença celíaca em crianças e adolescentes portadoras de baixa-estatura e pertencentes a famílias de baixa-renda aponta para a necessidade de investigação sistemática da doença celíaca nesses pacientes.
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Bhadada SK, Bhansali A, Kochhar R, Menon AS, Sinha SK, Dutta P, Nain CK. Does every short stature child need screening for celiac disease? J Gastroenterol Hepatol 2008; 23:e353-6. [PMID: 18086116 DOI: 10.1111/j.1440-1746.2007.05261.x] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/09/2022]
Abstract
AIM To study the etiological profile of short stature at a tertiary care hospital in north India. METHODS In this prospective study, 176 children with short stature were enrolled from January 2005 to December 2006. Appropriate screening and definitive tests were performed to establish the etiology of short stature. RESULTS Celiac disease (CD) emerged as the single most common (15.3%) cause of short stature, followed by various endocrine disorders. It was interesting to note that none of the CD patients presented with gastrointestinal symptoms. CONCLUSION All short children should be screened for CD irrespective of gastrointestinal symptoms.
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Affiliation(s)
- Sanjay Kumar Bhadada
- Departments of Endocrinology, Postgraduate Institute of Medical Education and Research Chandigarh, India
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Barreto AM, Bigolin MC, Ramos JCRR, Machado LPRR, Silva LDR, Silveira RBD, Boguszewski MCS. [Growth hormone therapy for children with chronic diseases]. ARQUIVOS BRASILEIROS DE ENDOCRINOLOGIA E METABOLOGIA 2008; 52:774-782. [PMID: 18797584 DOI: 10.1590/s0004-27302008000500009] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 05/01/2008] [Accepted: 05/20/2008] [Indexed: 05/26/2023]
Abstract
Growth disorders are commonly observed in children suffering from chronic diseases. The pathogenesis of growth failure is multifactorial. In chronic inflammatory diseases such as juvenile idiopathic arthritis and inflammatory bowel disease, growth is also affected by pro-inflammatory cytokines. Patients with chronic diseases might also become growth hormone (GH) deficient. However, normal or increased GH secretion with reduced plasma concentrations of insulin-like growth factor-I indicate a degree of GH insensitivity in some patients. Growth damage can increase with specific treatments, especially if glucocorticoids are used. GH therapy has been used to reduce the consequences of the disease and long-term steroid therapy in these patients. In this review, it is reported the encouraging results of GH treatment in growth-retarded children with chronic diseases, both in well defined indications as well in situations still under investigation.
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Affiliation(s)
- Alexandre M Barreto
- Unidade de Endocrinologia Pediátrica, Departamento de Pediatria, Universidade Federal do Paraná, Curitiba, PR, Brazil
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Growth hormone treatment in prepubertal children with celiac disease and growth hormone deficiency. J Pediatr Gastroenterol Nutr 2007; 45:433-7. [PMID: 18030209 DOI: 10.1097/mpg.0b013e3180de5e0a] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/14/2022]
Abstract
OBJECTIVE To evaluate the growth response to growth hormone (GH) replacement therapy during a gluten-free diet in patients with celiac disease (CD) associated with GH deficiency (GHD). PATIENTS AND METHODS A total of 14 prepubertal children affected by CD and GHD with no catch-up growth after >/=12 months of gluten-free diet and a reversion to seronegativity for antiendomysium antibodies and 10 age-matched prepubertal children with idiopathic GHD (IGHD) entered the study. All of the patients were treated with the same GH dosage (0.25 mg/kg subcutaneously each week). Height, growth rate, and body mass index were measured at the time of diagnosis of CD, at the time of endocrinological evaluation, and after the first, second, and third year of GH replacement therapy. RESULTS Growth rate strikingly increased (P < 0.005) during the first year of therapy in a similar way in subjects with CD/GHD and IGHD (from a median standard deviation score [SDS] of -2.34 to an SDS of 3.25 and from an SDS of -1.29 to an SDS of 2.79, respectively). During the second and third years of GH treatment, the growth rate tended to decrease but the values at the third year were always positive (CD/GHD, median SDS, 1.10; IGHD, median SDS, 0.11), indicating continued catch-up growth. CONCLUSIONS In patients with CD with GH deficiency confirmed after >/=12 months of gluten-free diet, GH replacement therapy should be started to allow complete catch-up growth in children. In addition, the effect of GH treatment in patients who comply with a gluten-free diet seems to be comparable to that observed in children with IGHD.
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