| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Rheumatology 2001; 40: 1196-1197
© 2001 British Society for Rheumatology
Letters to the Editor |
Sjögren's syndrome: a potential new aetiology of mild cobalamin deficiency
Departments of Internal Medicine and
1 Nuclear Medicine, Hôpital de Hautepierre, University Hospital of Strasbourg, France
SIR, Malabsorption of cobalamin from the food [FCM (food cobalamin malabsorption)] is one of the leading causes of cobalamin deficiency [1]. The scientific basis of FCM is the inability to isolate cobalamin from food or from its binders (particularly haptocorrin) without acid or pepsin [2]. Various conditions or disorders may be associated with FCM, mainly atrophic gastritis and partial gastric surgery [2]. In recent years, several other conditions or agents have been identified: stomach stapling (for obesity), acid-suppressive drugs (H2 antagonists, proton pump inhibitors) [3] and Helicobacter pylori infection [4]. In this letter, we report a new aetiology of FCM: Sjögren's syndrome.
During a 5-yr period (1995–2000), 127 patients with cobalamin deficiency (serum vitamin B12 level <200 pg/ml by radioimmunoassay [Bayer, New York, NY, USA]) were enrolled in a cohort study (partial data published in [1]). Eighty patients (63%) were identified as suffering from FCM (E. Andrès et al., unpublished data) but had a normal Schilling test result [using free cyanocobalamin labelled with cobalt-58 (Dicopac test; Amersham Healthcare, Birmingham, UK)], no anti-intrinsic factor antibodies and no dietary vitamin B12 deficiency [2].
Sjögren's syndrome fulfilling the European criteria [5] was found in three patients (3.75%), together with xerophthalmia and/or xerostomia and Chisholm grade 3 or 4 on salivary biopsy [5]. Baseline characteristics of these patients are listed in Table 1
. One patient (patient 2 in Table 1) had evidence of rheumatoid arthritis: destructive arthritis of the fingers and the carpus, rheumatoid factor [1/128 by enzyme-linked immunosorbent assay (ELISA)] and anti-Ro antibodies (1/32 by ELISA). Two of the three patients were treated with oral crystalline cyanocobalamin (500 µg/day); after 3 months of treatment, the cobalamin deficiency was corrected (mean serum vitamin B12 level 225 pg/ml) in both patients.
|
Our patients had mild cobalamin deficiency (mean serum vitamin B12 level 144 pg/ml) that was related to unquestionable FCM (using the criteria proposed by Carmel [2]) and Sjögren's syndrome (using the European criteria [5]). To our knowledge, a link between Sjögren's syndrome and FCM has not been reported previously; only a putative association between Sjögren's syndrome and pernicious anaemia (both of which are autoimmune disorders) has been suggested.
In our experience, Sjögren's syndrome is the cause of 3.75% of cases of FCM and 2.4% of all cases of cobalamin deficiency. The pathogenesis of this association is not known, but several mechanisms may be possible, such as a low haptocorrin level related to cellular immune infiltration of salivary glands (Chisholm grade 3 or 4 for our three patients), achlorhydria related to gastric infiltration (our three patients had gastritis with immune cellular infiltration) and/or mild pancreatic insufficiency related to Sjögren's syndrome [6]. It is important to note that an association of Sjögren's syndrome with pernicious anaemia may also occur in some cases of cobalamin deficiency [7].
In our opinion, it is important to be aware of the possibility of the association of FCM with Sjögren's syndrome, because recognition of this association will offer the possibility of preventing severe cobalamin deficiency easily (with oral vitamin B12 [8]) and of reducing the incidence of hyperhomocysteinaemia, which is associated with atherosclerosis and thrombotic events [9]. Moreover, the diagnosis of FCM enables one to propose therapy with oral vitamin B12 [8].
Notes
Correspondence to: E. Andrès, Service de Médecine Interne, Clinique Médicale B, 1 place de l'Hôpital, F67098 Strasbourg Cedex, France. 
References
-
Andrès E, Goichot B, Schlienger JL. Food-cobalamin malabsorption: a usual cause of vitamin B12 deficiency. Arch Intern Med2000;160:2061–2.
[Free Full Text] - Carmel R. Malabsorption of food-cobalamin. Baillière's Clin Haematol1995;8:639–55.[Web of Science][Medline]
-
Marcuard SP, Albernaz L, Khazani PG. Omeprazole therapy causes malabsorption of cyanocobalamin (vitamin B12). Ann Intern Med1994;120:211–5.
[Abstract/Free Full Text] -
Kaptan K, Beyan C, Ural AU, Cetin T, Avcu F, Gülsen M et al. Helicobacter pylori. Is it a novel causative agent in vitamin B12 deficiency? Arch Intern Med2000;160:1349–53.
[Abstract/Free Full Text] - Kaplan G. Syndrome de Gougerot–Sjögren. In: Kahn MF, Peltier AP, Meyer O, Piette JC, eds. Les maladies systémiques, edn. 3. Paris: Flammarion Médecine-Sciences, 1991:499–544.
- Sheikh SH, Shaw-Stiffel TA. The gastrointestinal manifestations of Sjögren's syndrome. Am J Gastroenterol1995;90:9–14.[Medline]
- Andrès E, Perrin AE, Kraemer JP, Goichot B, Demengeat C, Ruellan A et al. Anémies par carence en vitamine B12 chez le sujet âgé de plus de 75 ans: nouveaux concepts. A propos de 20 observations. Rev Med Intern2000;21:946–55.
-
Kuzminski AM, Del Giacco EJ, Allen RH, Stabler SP, Lindenbaum J. Effective treatment of cobalamin deficiency with oral cobalamin. Blood1998;92:1191–8.
[Abstract/Free Full Text] - Refsum H, Ueland PM, Nygard O, Vollset SE. Homocysteine and cardiovascular disease. Annu Rev Med1998;49:31–62.[Web of Science][Medline]
CiteULike 
Connotea 
Del.icio.us What's this?
This article has been cited by other articles:
|
|
 |
|
  N. Dali-Youcef and E. Andres An update on cobalamin deficiency in adults QJM, January 1, 2009; 102(1): 17 - 28. [Abstract] [Full Text] [PDF]
|
|
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||