International Journal of Clinical Case Reports 2017, Vol.7, No.1, 1-3
2
1997)), Hashimoto thyroiditis and celiac disease. Besides a gluten free diet, the patient was treated with
hydroxychloroquine (400 mg/day) and L-thyroxine (100 µg/day). She responded positively to this treatment and is
being regularly followed up in our outpatient clinic. Ten years later, no relapse was noted.
2 Discussion
Autoimmune diseases encompass a wide spectrum of diseases, from organ specific Hashimoto's thyroiditis to
systemic diseases such as systemic lupus erythematosus. Although the etiology is still poorly understood, genetic,
immunological, hormonal and environmental factors are the major predisposing and triggering factors
(Szyper-Kravitzm et al., 2005). Studies documenting increased risk of developing autoimmune diseases (ADs)
have shown that these conditions share several immunogenetic mechanisms (Koning, 2015). HLA A1B8DR3
haplotype is associated to celiac disease (Castiblanco et al., 2015). This haplotype is seen in systemic lupus
erythematosus in 40 to 70% of cases (Sparks and Costenbader, 2014). Hence, the two diseases can occur in the
same patient as they share common genetic background. The multiple autoimmune syndrome were described by
Humbert and Dupond in 1988 as a syndrome(s) consisting of the presence of three or more well characterized
ADs in one patient (Humbert and Dupond, 1988). This definition is based on 91 reported cases of such
associations in the literature (Mohan and Ramesh, 2003). MAS can be classified into three groups according to the
prevalence of their associations with one another. Type 1 MAS includes myasthenia gravis, thymoma,
polymyositis, and giant cell myocarditis. Type 2 includes Sjögren's syndrome, rheumatoid arthritis, primary biliary
cirrhosis, scleroderma, and autoimmune thyroid disease. Type 3 groups together autoimmune thyroid disease,
myasthenia gravis and/or thymoma, Sjögren's syndrome, pernicious anaemia, idiopathic thrombocytopenic
purpura, Addison's disease, insulin-dependent diabetes, vitiligo, autoimmune haemolytic anaemia, SLE, and
dermatitis herpetiformis (Mohan and Ramesh, 2003). Systemic lupus erythematosus (SLE), autoimmune thyroid
disease (AITD), and Sjögren's syndrome together were the most frequent ADs encountered (Anaya et al., 2012).
For our case, with the presence of Hashimoto’s thyroiditis and SLE, our patient seems to qualify, but only partially,
with type 3 MAS. Celiac disease, though an immune mediated disorder, is not included in the classification of
MAS and usually associated to type 1 diabetes mellitus (Zhao et al., 2016). The review of literature revealed only
one case report with these conditions occurring in a pediatrician patient although there have been reports of
variable association between any two of these conditions. In Fact, Latif et al reported the case of an 11 year-old
patient presenting general symptoms, constipation, joint pain, and goiter. The final diagnosis was MAS. NSAIDS
with short course of corticosteroids were given for SLE, levothyroxine for Hashimoto’s thyroiditis, and gluten free
diet was advised for coeliac disease. The patient responded positively to this treatment (Latif et al., 2010). A
patient suffering from one autoimmune disease has 25% chances of acquiring another autoimmune disease
(Mohan and Ramesh, 2003). The association of celiac disease and systemic lupus erythematosus is uncommon.
Few cases were reported (Zitouni et al., 2004; Hrycek and Siekiera, 2008; Freeman et al., 2008). In a case series
of 30 patients affected with MAS, celiac disease was associated to Hashimoto’s thyroiditis in 9 patients (Giardino
et al., 2011). This association is frequent and well known in the literature with as prevalence estimated at 5.4%.
This could be explained by the sharing of a common genetic predisposition by the two disorders, namely the DQ2
allele (Kumar et al., 2001). The association of Hashimoto’s thyroiditis with systemic lupus erythematosus has
been reported in adults in a range of 7.5% to 8.9% (Zitouni et al., 2004).
3 Conclusions
According to definition; the combination of three autoimmune diseases in our patient qualifies for the diagnosis of
MAS. This case illustrates the importance of regular follow-up of each patient with autoimmune disease, in order
to detect other associated autoimmune conditions. Genetics background plays an important part in its occurrence.
Authors’ contributions
Nabil Belfeki and Souheil Zayet followed the patient at the outpatient clinic. Nabil Belfeki and Imed Ben Ghorbel performed the
bibliography for this case. Mohamed Habib Houman supervised the whole case before its submission in International Journal of
Clinical Case Reports. All authors read and approved the final manuscript.
