Dear Label_Name,

Greetings! I hope you are well.

I am writing on behalf of an international publishing house, Science and Education Publishing, USA (http://www.sciepub.com).

Science and Education Publishing (SciEP) is a multidisciplinary open access publisher of Journals covering the fields of Science, Technology and Medicine. All SciEP publications are subject to high-quality peer review, editorial and production processes and are freely available online.

Welcome to the April, 2013 edition of SciEP newsletter, providing monthly summaries of articles on science & education related topics of interest to scientist and professionals. The mission of our journals is to provide an open access forum for publishing new findings on issues pertinent to scientific and academic field. We also invite you to submit original research papers, review articles, short reports, or case studies are considered for publication in our Multidisciplinary Journals.

American Journal of Public Health Research
DOI: 10.12691/ajphr-1-1-3

The Implementation of a Tele-Homecare System with Service Oriented Architecture and HL7 Message Transmission Standard
Ching-Sung Wang

American Journal of Medical and Biological Research
DOI: 10.12691/ajmbr-1-1-2

The Influence of Ethanol on Pyruvate Kinases Activity in Vivo, in Vitro, in Silico
Lelevich Sergey Vladimirovich, Khrustalev Vladislav Victorovich, Barkovsky Eugene Victorovich, Shedogubova Tatyana Aleksandrovna

American Journal of Medical Sciences and Medicine
DOI: 10.12691/ajmsm-1-1-1

Clinical and Histological Features in Wegener Granulomatosis
Li Cavoli Gioacchino, Passantino Rita, Bono Luisa, Tortorici Calogera, Ferrantelli Angelo, Giammarresi Carlo, Rotolo Ugo

American Journal of Infectious Diseases and Microbiology
DOI: 10.12691/ajidm-1-1-2

The Curse of Lymphatic Filariasis: Would the Continual Use of Diethylcarbamazine Eliminate this Scourge in Papua New Guinea?
Anne Lanham, Lillian Mwanri

We have released issues of some journals on February 2013.
American Journal of Public Health Research
American Journal of Medical Sciences and Medicine
American Journal of Medical and Biological Research
American Journal of Pharmacological Sciences
American Journal of Clinical Medicine Research
American Journal of Nursing Research
American Journal of Cancer Prevention
American Journal of Medicine Studies
American Journal of Infectious Diseases and Microbiology
American Journal of Cardiovascular Disease Research
American Journal of Biomedical Research
American Journal of Mechanical Engineering
American Journal of Microbiological Research
Journal of Preventive Medicine
American Journal of Food and Nutrition
American Journal of Hypertension Research
American Journal of Sports Science and Medicine
Journal of Cancer Research and Treatment
American Journal of Zoological Research
Journal of Biomedical Engineering and Technology
American Journal of Microbiological Research
Biomedical Science and Engineering

We are pleased to announce that submissions are now being accepted for peer review for all the journals across all our subject areas in SciEP.

Please submit your manuscript via our online submission system:
http://www.sciepub.com/author/signin

To unsubscribe from our mailing list, please click on the following link: unsubscribe

I am looking forward to hearing from you.

Kind regards,

David Rain
Email:join@sciepub.com
Science and Education Publishing (http://www.sciepub.com)

Pleuro-pericarditis Revealing Celiac Disease

Wafa Ben Saada, Fatma Derbali*, Nawrez Kammoun, Raouf Hajji, Sana Triki, Monia Elleuch, Saida Zribi, Mohammed Jallali, Rim Masmoudi

Internal Medecine Department; Regional Hospital of Sidi Bouzid; Tunisia

Abstract: Background: Celiac disease (CD) is an auto immune disease that often one or more disorders may be associated especially endocrine but also heart disorders. We report a patient case with idiopathic hypo-parathyroidism and CD revealed by pleuro-pericarditis. Observation: A 23-year-old woman was admitted in intensive care unit for epileptic-evil condition following dyspneising pleurisy. She has been diagnosed as epileptic case since 5 years and she had undergone surgery for right cataract. On the physical examination, she exhibited a tachycardia and breathing rales at pulmonary auscultation. She had clinical signs of chronic hypocalcaemia, without gastrointestinal complaints. Chest x-ray revealed cardiomegaly and right pleurisy. The cardiac ultrasound demonstrated very abundant bilateral pleural and pericardial effusion. The phosphocalcic balance resulted in a severe hypocalcemia at 0.8 mmol / l and hypo-parathyroidism was confirmed. In addition to the surgical drainage, she had calcium and vitamin D supplementation. CD was suspected after the resistance for a high supplementation and confirmed by endoscopy. Under a gluten-free diet (GFD), the evolution was marked by the stability of serum calcium levels and disappearance of pleural and pericardial effusion. Conclusion: The possibility of celiac disease should be considered in patients with hypoparathyroidism and pericarditis that seems unduly difficult to treat. This should be evaluated even in the absence of gastrointestinal symptoms.

Keywords: hypocalcoemia, malabsorption syndrome, pleuro-pericarditis, hypoparathyroidism, celiac disease

Citation: Wafa Ben Saada, Fatma Derbali, Nawrez Kammoun, Raouf Hajji, Sana Triki, Monia Elleuch, Saida Zribi, Mohammed Jallali, Rim Masmoudi, Pleuro-pericarditis Revealing Celiac Disease. International Journal of Celiac Disease, 5 (1). 35-39. January 2017. doi:10.12691/ijcd-5-1-10

1. Introduction

Celiac disease (CD) is an auto immune small intestinal mucosal disorder that occurs in genetically predisposed individuals as the result of an immune response to gluten. It is present in approximately 1% of the population [1]. Diarrhea has become a less common mode of presentation (<50% of cases) than it once was [2]. During the last decades CD has increasingly become recognized as a multi-organ disorder, and has been linked to a number of complications including malignancy [3], autoimmune disorders [4,5] and tuberculosis [6]. Individuals with CD are also at increased risk of death from ischemic heart disease [7]. To our knowledge, research on CD and pericarditis is limited to case-reports [8,9,10,11,12].

We report the clinical case of an association between idiopathic hypoparathyroidism (IH) and a celiac disease revealed by pleuro-pericarditis.

2. Case Report

A 23-year-old woman with a history of epilepsy for 5 years and cataract of the right eye operated 3 years ago, admitted in intensive care unit for epileptic-evil condition. The clinical examination found a tachycardia at 104 beats/min, a blood pressure at 10/4cmHg. Endocrinological examination was normal. The inflammatory assessment revealed hyper-leucocytosis at 22,700 mmol/l. Chest x-ray revealed cardiomegaly and right pleurisy. The electro-cardiogram found diffuse microvoltage and flattening T-wave. A cardiac ultrasound demonstrating a bilateral pleural effusion and abundant pericardial with signs of pre-tamponade. The patient had pericardial and pleural surgical drainage. Bacteriological, mycological, immunological and tuberculosis investigations were negative. The patient was treated by colchicine 1 mg/day and aspegic 100 mg / day. The cardiac echocardiography realized at 1 week after surgery demonstrated a very low abundance pleural and pericardial effusion.

On physical examination, we noticed chronic hypocalcaemia clinical signs (muscle cramps, cataract of the right eye). The biological analysis showed a severe hypocalcemia (0.8 mmol / l). The parathyroid hormone was less than 3 pg / ml which allowed to retain the diagnosis hypo-parathyroidism.

There was no improvement in serum calcium levels after oral calcium and vitamin D (calciferol) supplementation on reversal of calcium parenteral supplementation (Figure 2). Thus celiac disease has been suspected. The patient had no chronic diarrhea but biological malabsorption signs (hypo-calcemia, anemia, hypo-albuminemia and hypocholesterolemia). Anti-transglutaminase antibodies were positive. Oeso-gastroduodenal fibroscopy confirmed villous atrophy. The patient followed a Gluten Free Diet (GFD) and had an oral calcium and vitamin D supplementation. The evolution was marked by an improvement in the biological malabsorption signs: disappearance of anemia, rise in blood protein and calcium levels (Figure 1) and disappearance of pleural and pericardial effusion (Figure 2a and Figure 2b).

Figure 1. Evolution of calcium and phosphoric levels under treatment

Figure 2. a: Chest x- ray before GFD: Pleuro-pericarditis, b: Chest x ray after GFD: disappearance of Pleuro-pericarditis

3. Discussion

During the last decades CD has increasingly become recognized as a multi-organ disorder including autoimmune disorders [4,5] and cardiovascular diseases [11].

Several study showed that patients with CD are also at increased risk of ischemic heart disease [13], atrial fibrillation [14], cardiovascular death [15], dilated cardiomyopathy [16], autoimmune myocarditis and pericarditis [7,17]. All these studies were however, based on few positive events and with the exception of the prospective Danish study [18], they were cross-sectional and did not take into account whether CD preceded or ensued heart disease [19]. However, research on CD and pericarditis is limited to case-reports [8,9,10,11,12].

The majority of cases of pericarditis are idiopathic [20]. A viral etiology is often implicated but rarely proven [20]. In some cases of pericarditis, particularly when recurrent, autoimmune or hypersensitivity states have been implicated. This may be relevant to celiac disease where an association with immune disturbance is recognized [20].

In this context, pericarditis has previously been reported in CD, and it may even manifest as a primary presenting feature [21]. The likely mechanism is a disturbed humoral and cell-mediated immunity including other autoimmune reactions. There is deposition of circulating immune complexes originating from the small intestine, which may resemble the etiology of pericarditis similar to the one encountered in serum sickness [12,21]. The mechanisms are likely related to pericardial deposition of soluble antigen-antibody complexes when there is excess of antigen [20,21].

However, in the cohort study conducted by Elfström et al, they found no statistically significant association between CD and having later cardiomyopathy, pericarditis or myocarditis, but the risk estimated for pericarditis in CD were statistically significantly higher in CD diagnosed in adulthood compared with CD diagnosed in childhood [19].

Riccabona in his study reported that 50% of children with celiac disease had asymptomatic and limited pericardial effusion that only detectable with instrument [22]. Anemia is the most common presentation of CD in adults and it may be the only presentation of disease and iron deficiency is the major cause of this anemia [23]. Anemia and leg edema were the major findings and pericardial effusion was asymptomatic in the case reported by Farzaneh [11]. In our patient, anemia was not severe, pericardial effusion was not symptomatic and it was an incidental finding; and the diagnosis of CD was based on serologic and histologic findings.

After appropriately excluding other autoimmune conditions and if pericardial symptoms of CD patients do not completely respond to GFD alone, several authors recommended steroids as a first-line therapy at least for a while [20,24]. It is in contrast to the treatment of acute idiopathic pericarditis where one would consider either non-steroidal anti-inflammatory drugs or colchicine [21,24]. Colchicine has not been extensively studied in patients with autoimmune diseases; however at least 7% of the patients in CORP-2 trial had underlying autoimmune conditions and benefited from the use of colchicine [25]. Our patient had a pericardial surgical drainage followed by treatment with colchicine and aspegic and she followed a GFD.

However, the intestine is one of the most involved organs in adverse drug reactions (20 – 40%) [26]. The entero-toxicity may be an immediate but also delayed effect. Colchicine can essentially lead to diarrhea. Isolated cases of villous atrophy were attributed to mefenamic acid, azathioprine, methotrexate, ticlopidine and recently to mycophenolate and olmesartan [27]. This can’t be the case for our patient. The fact that the patient didn’t take anti-inflammatory drugs nor colchicine before and the positivity of anti-transglutaminase antibodies and the favorable clinical evolution under GFD are against the diagnosis of drug enteropathy.

On the other hand, IH can be associated with other digestive autoimmune diseases that may cause diarrhea [28]. Cases of IH coexisting with CD are described in a handful of cases in the literature, [29,30] although this situation occurs very rarely and mainly in patients with long standing disease and therefore lengthy exposure to antibodies [31]. The described association could be an occasional finding, but a common pathogenesis of both diseases can be hypothesized on immunological basis [30]. Studies have found that parathyroid antibodies can be of two types: one specific for the parathyroid gland, the other non- specific and able to mimic the anti-endomysial antibodies associated with CD. Besides, tissue transglutaminase antibodies produced in CD have been shown to cross-react with other antigens and endomysial antibody reactions on parathyroid tissue have been shown to lead to parathyroid atrophy [31,32,33]. Soma et al. assessed in their comparative cohort study that the prevalence of CD and anti- tissue transglutaminase autoantibodies (anti-tTGAbs) in IH was 6,4% [34]. The age of the disease onset ranged from early childhood to old age [30].

Hypoparathyroidism is a chronic disorder characterized by hypocalcaemia, tetany, cataract, basal ganglia calcification and convulsions [35,36]. Our patient was 23 years old and she had probably chronic hypocalcaemia that can be involved in recurrent cataract and in the epilepsy.

In some reports, hypoparathyroidism was diagnosed some years prior to the diagnosis of CD [30], while in other reports, the diagnosis of hypoparathyroidism was made concomitantly with the diagnosis of CD [37]; such as in our case report.

Hypoparathyroidism causes hypocalcaemia through a fall in parathyroid hormone levels, leading to reductions in bone reabsorption and intestinal calcium absorption and, in patients without end-stage renal disease, increased calcium excretion. In CD, the mechanism leading to hypocalcaemia is chronic inflammation of the intestinal mucosa leading to malabsorption of vitamin D and calcium [38]. Hypocalcaemia is often exacerbated by a decreased intake of dairy products because of lactose intolerance, as lactase cannot be produced by degenerated intestinal epithelial cells and is not available to cleave the lactose in dairy products [39].

Hypocalcaemia may present as an asymptomatic laboratory finding or as a severe, life-threatening condition. Upon the diagnosis of acute hypocalcaemia, a rapid treatment regimen may be necessary. In contrast, chronic hypocalcaemia may be well tolerated by the patient with hypoparathyroidism like the case of our patient, but treatment nonetheless remains necessary in order to prevent long-term complications [37].

If suspected by resistance to vitamin D supplementation [40], the coexistence of CD must be ruled out by duodenal biopsy. In such cases, GFD should be included in the treatment regimen [30,41]. Once a GFD is adhered to, approximately 70% of patients notice symptomatic improvement within two weeks, anti-endomysial antibodies generally disappear in 6–12 months and plasma calcium levels normalize quickly [42], as illustrated also in our case report.

Many other studies also assessed the effect of a GFD on calcemic control in patients with IH, found to have CD. A GFD led to improvement in weight and calcium control during follow-up in patients with hypoparathyroidism and CD [34,37,41,43]. In fact, in some cases, hypercalcemia was noted after the institution of a GFD, necessitating a reduction in the daily dose of calcium and vitamin D therapy [42].

However, despite dietary compliance, the speed and degree of histologic improvement is unpredictable but invariably lags behind the clinical response [42]. Data indicates that mucosal inflammation may persist for many years and histologic improvement may not be evident on repeated biopsy for 2–3 months [42].

4. Conclusion

Our observation is particular by the simultaneous diagnosis of hypoparathyroidism and CD following severe pleuro-pericarditis.

The possibility of CD should be considered in patients with hypoparathyroidism that seems unduly difficult to treat. This should be evaluated even in the absence of gastrointestinal symptoms.

So it is important to be aware of a potential co-occurrence of CD in patients with IH, wherein a GFD has beneficial impact on calcium control.

Besides, in such association, a long-term monitoring is necessary to detect recurrence of pericarditis.

References

[1]   | Fasano A, Berti I, Gerarduzzi T et al, “Prevalence of celiac disease in at-risk and not-at-risk group in the United States: a large multicenter study,” Arch Intern Med 2003, 163: 286-92.
[2]   | Hernandez L, Green PH, “Extra-intestinal manifestations of celiac disease,” Curr Gastroenterol Rep, 2006, 8:383-9.
[3]   | Askling J, Linet M, Gridley G, Halstensen TS, Ekstrom K, Ekbom A, “Cancer incidence in a population-based cohort of individuals hospitalized with celiac disease or dermatitis herpeti-formis,” Gastroenterology 2002, 123: 1428-35.
[4]   | Ventura A, Magazzu G, Greco L, “Duration of exposure to gluten and risk for autoimmune disorders in patients with celiac disease. SIGEP Study Group for Autoimmune Disorders in Celiac Disease,” Gastroenterology 1999, 117: 297-303.
[5]   | Ludvigsson JF, Ludvigsson J, Ekbom A, Montgomery SM, “Celiac disease and risk of subsequent type 1 diabetes: A general population cohort study of children and adolescents,” Diabetes Care 2006, 29: 2483-8.
[6]   | Ludvigsson JF, Wahlstrom J, Grunewald J, Ekbom A, Montgomery SM, “Coeliac disease and risk of tuberculosis: a population based cohort study,” Thorax 2007, 62: 23-8.
[7]   | Peters U, Askling J, Gridley G, Ekbom A, Linet , “Causes of death in patients with celiac disease in a population-based Swedish cohort,” Arch Intern Med 2003, 163: 1566-72.
[8]   | Faizallah R, Costello FC, Lee FI, Walker R, “Adult celiac disease and recurrent pericarditis,” Dig Dis Sci 1982, 27: 728-30.
[9]   | Laine LA, Holt KM, “Recurrent pericarditis and celiac disease,” JAMA 1984, 252: 316.
[10]   | P. Elfstrom et al, “Coeliac disease and heart disease,” Blackwell Publishing Ltd Journal of Internal Medicine 2007, 262: 545-554.
[11]   | Farzaneh A, Ali D, Mitra H, Tahmineh T, and Jamshid N. “Pericardial Effusion in Celiac Disease” Int J Prev Med. 2014, 5: 356-359.
[12]   | PT Dawas, ST Atherton, “Coeliac disease presenting as recurrent pericarditis” The Lancet. 1981, 1021-1022P. T. DAWES S.
[13]   | Ludvigsson JF, James S, Askling J, Stenestrand U, Ingelsson E. “Nationwide cohort study of risk of ischemic heart disease in patients with celiac disease.” Circulation. 2011, 123: 483-90.
[14]   | Emilsson L, Smith JG, West J, Melander O, Ludvigsson JF. “Increased risk of atrial fibrillation in patients with coeliac disease: A nationwide cohort study.” Eur Heart J. 2011, 32: 2430-7.
[15]   | Ludvigsson JF, Montgomery SM, Ekbom A, Brandt L, Granath F. “Small-intestinal histopathology and mortality risk in celiac disease.” JAMA. 2009, 302: 1171-8.
[16]   | Emilsson L, Andersson B, Elfström P, Green PH, Ludvigsson JF. “Risk of idiopathic dilated cardiomyopathy in 29 000 patients with celiac disease.” J Am Heart Assoc. 2012, 1: e001594.
[17]   | Frustaci A, Cuoco L, Chimenti C, Pieroni M, Fioravanti G, Gentiloni N, et al. “Celiac disease associated with autoimmune myocarditis.” Circulation. 2002, 105: 2611-8.
[18]   | Fonager K, Sorensen HT, Norgard B, Thulstrup AM. “Cardiomyopathy in Danish patients with coeliac disease.” Lancet 1999, 354: 1561.
[19]   | Elfström P, Hamsten A, Montgomery SM, Ekbom A, Ludvigsson JF. “Cardiomyopathy, pericarditis and myocarditis in a population-based cohort of inpatients withcoeliac disease.” J Intern Med. 2007, 262: 545-54.
[20]   | Tzivoni D, Licht A, Eliakim M: “Acute idiopathic pericardity in the aged.” Eur J Cardiol 1975, 2/3:327-335.
[21]   | Lovely Ch, Nauman Kh, and David H. S, “Pericardial Manifestations in Autoimmune Encounters: Some Worthy Facts for Consideration!” Int J Prev Med. 2014, 5: 1632-3.
[22]   | Riccabona M, Rossipal E. “Pericardial effusion in celiac disease: An incidental finding?” Wien Klin Wochenschr. 2000, 112: 27-31.
[23]   | Lo W, Sano K, Lebwohl B, Diamond B, Green PH. “Changing presentation of adult celiac disease.” Dig Dis Sci. 2003; 48: 395-8.
[24]   | Chhabra L, Spodick DH. Letter by Chhabra and Spodick regarding article, “Treatment of acute and recurrent idiopathic pericarditis” Circulation. 2013, 128-391.
[25]   | Imazio M, Belli R, Brucato A, Cemin R, Ferrua S, Beqaraj F, et al. “Efficacy and safety of colchicine for treatment of multiple recurrences of pericarditis (CORP-2): A multicentre, double-blind, placebo-controlled, randomised trial.” Lancet. 2014, 383: 2232-7.
[26]   | Chassany O, Michaux A, Bergmann JF. “Drug induced diarrhea.” Drug Safety 2000, 22: 53-72.
[27]   | Alexandre Pariente “Entéropathies médicamenteuses ” POST’U, 2015. [E-book] Available: http://www.fmcgastro.org/ 221_228_Pariente.pdf.
[28]   | Abboud B et al. “Digestive manifestations of dysparathyroidism” World J Gastroenterol 2011,17: 4063-6.
[29]   | Khandwala HM, Chibbar R, Bedia A. “Celiac disease occurring in a patient with hypoparathyroidism and autoimmune thyroid disease.” South Med J 2006, 99: 290-2.
[30]   | Isaia GC, Casalis S, Grosso I, Molinatti PA, Tamone C, Sategna-Guidetti CJ. “Hypoparathyroidism and co-existing celiac disease.” J Endocrinol Invest 2004, 27: 778-81.
[31]   | Kumar V, Valeski JE,Wortsman J. “Celiac disease and hypoparathyroidism: cross-reaction of endomysial antibodies with parathyroid tissue.” Clin Diagn Lab Immunol 1996, 3: 143-6.
[32]   | Kumar V, Rajadhyaksha M,Wortsman J. “Celiac disease associated autoimmune endocrinopathies.” Clin Diagn Lab Immunol 2001, 8: 678-85.
[33]   | Baruch Y, Raffi L-T and Neta L “Refractory Hypoparathyroidism in a Child with Celiac Disease” IMAJ 2016, 18: 58-60.
[34]   | Saha S, Saini S, Makharia GK, Gupta SD, Goswami R “Prevalence of coeliac disease in idiopathic hypoparathyroidism and effect of gluten-free diet on calcaemic control” Clin Endocrinol (Oxf) 2016, 84, 578-586.
[35]   | Shoback, D. “Clinical practice. Hypoparathyroidism” N Engl J Med 2008, 359, 391-403.
[36]   | Bilezikian, J.P., Khan, A., Potts, J.T. Jr et al. “Hypoparathyroidism in the adult: epidemiology, diagnosis, pathophysiology, target-organ involvement, treatment, and challenges for future research.” J Bone Miner Res, 2011, 26: 2317-2337.
[37]   | José A, Miguel M, Pedro S J, Cristina A, Proques da Silveira M. “Resistance to vitamin D treatment as an indication of celiac disease in a patient with primary hypoparathyroidism” CLINICS 2009, 64: 259-61.
[38]   | Van Heel DA, West J. “Recent advances in coeliac disease.” Gut 2000, 55: 1037-46.
[39]   | Selby PL, Davies M, Adams JE, Mawer EB. “Bone loss in celiac disease is related to secondary hypoparathyroidism” J Bone Miner Res 1999, 14: 652-7.
[40]   | Frysák Z, Hrcková Y, Rolinc Z, Hermanová Z, Lukl J. “Idiopathic hypoparathyroidism with celiac disease: diagnostic and therapeutic problem” Vnitr Lek. 2000, 46: 408-12.
[41]   | Sari R, Yildirim B, Sevinc A, Buyukberber S. “Idiopathic hypoparathyroidism and celiac disease in two patients with previous history of cataract” Indian J Gastroenterol. 2000, 19: 31-2.
[42]   | Sibylle Kohler, John AH Wass “ Hypoparathyroidism and coeliac disease: a potentially dangerous Combination” J R Soc Med 2009, 102: 311-314.
[43]   | Kara, B. & Sandikci, M. “Successful treatment of portal hypertension and hypoparathyroidism with a gluten-free diet.” J Clin Gastroenterol 2007, 41: 724-725.