Rheumatology International

, Volume 26, Issue 3, pp 261–263 | Cite as

Severe osteomalacia due to undiagnosed cœliac disease: three case reports of Tunisian women

  • Hanène Landolsi
  • Elyès Bouajina
  • Amani Mankaï
  • Hela Zeglaoui
  • Karim Skandrani
  • Ibtissem Ghedira
Case Report

Abstract

We describe three cases of osteomalacia presenting in Tunisian women, all of whom had previously-undiagnosed cœliac disease (CD). Direct enquiry revealed an important weight loss and a history of diarrhoea in two patients, and a 15-year history of anaemia in one patient. Laboratory tests showed severe anaemia in the three cases. Reduced calcium was found in two cases, and corrected calcium was found in one case. Radiological examination showed fissure in two cases. The diagnosis of osteomalacia was made by clinical, biochemical and radiological features. Antigliadin, antireticulin, antiendomysial and anti-tissue transglutaminase antibodies were all positive in the three cases, and a small-bowel biopsy confirmed the diagnosis of CD. Treatment with gluten-free diet (GFD), supplemental calcium and vitamin D was initiated for the three patients, but only one patient complies strictly with the GFD; she showed a marked resolution of her symptoms.

Keywords

Cœliac disease Osteomalacia 

Abbreviations

AEA

Antiendomysial antibodies

AGA

Antigliadin antibodies

ARA

Antireticulin antibodies

AtTGA

Anti-tissue transglutaminase antibodies

BMD

Bone mineral density

CD

Cœliac disease

GFD

Gluten-free diet

IEL

Increased intraepithelial lymphocytes

References

  1. 1.
    Kavak US, Yüce A, Koçak N, Demir H, Saltik IN, Gürakan F, Özen H (2003) Bone mineral density in children with untreated and treated celiac disease. J Pediatr Gastroenterol Nutr 37:434–436PubMedGoogle Scholar
  2. 2.
    Masala S, Annibale B, Fiori R, Capurso G, Marinetti A, Simonetti G (2003) DXA vs QCT for subclinical celiac disease patients. Acta Diabetol 40:S174–S176Google Scholar
  3. 3.
    Mustalahti K, Collin P, Sievänen H, Salmi J, Mäki M (1999) Osteopenia in patients with clinically silent cœliac disease warrants screening. Lancet 354:744–745CrossRefPubMedGoogle Scholar
  4. 4.
    Cellier C, Flobert C, Cormier C, Roux C, Schmitz J (2000) Severe osteopenia in symptom-free adults with a childhood diagnosis of celiac disease. Lancet 355:806CrossRefPubMedGoogle Scholar
  5. 5.
    De Boer WA, Tytgat GN (1992) A patient with osteomalacia as single presenting symptom of gluten sensitive enteropathy. J Intern Med 232:81–85PubMedGoogle Scholar
  6. 6.
    Lupattelli G, Fuscaldo G, Castellucci G, Ciuffetti G, Pelli MA, Mannarino E (1994) Severe osteomalacia due to gluten-sensitive enteropathy. Ann Ital Med Int 9:40–43PubMedGoogle Scholar
  7. 7.
    Russel JA (1994) Osteomalacic myopathy. Muscle Nerve 17:578–580CrossRefPubMedGoogle Scholar
  8. 8.
    Monti MA, Negri E, Paina S, Secchi GC (1996) Diagnosis of celiac disease in an elderly subject: description of a clinical case. Ann Ital Med Int 11:284–287PubMedGoogle Scholar
  9. 9.
    Dorst AJ, Ringe JD (1998) Severe osteomalacia in endemic sprue. An important differential diagnosis in osteoporosis. Fortschr Med 116:42–45Google Scholar
  10. 10.
    Gannage MH, Abikaram G, Nasr F, Awada H (1998) Osteomalacia secondary to celiac disease, primary hyperparathyroidism, and Graves’ disease. Am J Med Sci 315:136–139CrossRefPubMedGoogle Scholar
  11. 11.
    Basu RA, Elmer K, Babu A, Kelly CA (2000) Cœliac disease can still present with osteomalacia! Rheumatology 39:335–336CrossRefGoogle Scholar
  12. 12.
    García-Porrúa C, González-Gay MA, Avila-Alvarenga S, Rivas MJ, Soilan J, Penedo M (2000) Cœliac disease and osteomalacia: an association still present in Western countries. Rheumatology 39:1435CrossRefGoogle Scholar
  13. 13.
    Jensen ON, Reinholdt S, Ozdemir Z (2000) Overlooked celiac disease in a 59-year-old woman with severe osteomalacia! Ugeskr Laeger 162:670–671PubMedGoogle Scholar
  14. 14.
    Hepburn AL, Kaye SA (2001) Oligoarthritis in an elderly woman with diarrhoea and weight loss. Postgrad Med J 77:475–477CrossRefPubMedGoogle Scholar
  15. 15.
    Byrne MF, Abdul Razak AR, Leader MB, Sheehan KM, Patchett SE (2002) Disabling osteomalacic myopathy as the only presenting feature of cœliac disease. Eur J Gastroenterol Hepatol 14:1271–1274CrossRefPubMedGoogle Scholar
  16. 16.
    Wong M, Scally J, Watson K, Best J (2002) Proximal myopathy and bone pain as the presenting features of cœliac disease. Ann Rheum Dis 61:87–88Google Scholar
  17. 17.
    Moreno ML, Vazquez H, Mazure R, Smecuol E, Niveloni S, Perdreira S, Sugai E, Maurino E, Gomez JC, Bai JC (2004) Stratification of bone fracture risk in patients with celiac disease. Clin Gastroenterol Hepatol 2:127–134CrossRefPubMedGoogle Scholar
  18. 18.
    Vestergaard P (2003) Bone loss associated with gastrointestinal disease: prevalence and pathogenesis. Eur J Gastroenterol Hepatol 15:851–856PubMedGoogle Scholar
  19. 19.
    Hin H, Bird G, Fisher P, Mahy N, Jewell D (1999) Celiac disease in primary care: case finding study. BMJ 7177:164–167Google Scholar
  20. 20.
    Tursi A, Giorgetti G, Brandimarte G, Rubino E, Lombardi D, Gasbarrini G (2001) Prevalence and clinical presentation of subclinical/silent celiac disease in adults: an analysis on a 12-year observation. Hepatogastroenterology 48:462–464PubMedGoogle Scholar
  21. 21.
    Barera G, Mora S, Brambilla P, Ricotti A, Menn L, Beccio S, Bianchi C (2000) Body composition in children with celiac disease and the effects of a gluten-free diet: a prospective case-control study. Am J Clin Nutr 72:71–75PubMedGoogle Scholar
  22. 22.
    Ghedira I, Sghiri R, Ayadi A, Sfar MT, Harbi A, Essoussi AS, Amri F, Korbi S, Jeddi M (2001) Anticorps anti-endomysium, anti-réticuline et anti-gliadine, intérêt dans le diagnostic de la maladie cœliaque chez l’enfant. Pathol Biol (Paris) 49:47–52Google Scholar
  23. 23.
    Mankaï A, Sakly W, Landolsi H, Gueddah L, Sriha B, Ayadi A, Sfar MT, Skandrani K, Harbi A, Essoussi AS, Korbi S, Fabien N, Jeddi M, Ghedira I (2005) Tissue transglutaminase antibodies in celiac disease, comparison of an enzyme linked immunosorbent assay and a dot blot assay. Pathol Biol (Paris) 53:204–209Google Scholar

Copyright information

© Springer-Verlag 2005

Authors and Affiliations

  • Hanène Landolsi
    • 1
  • Elyès Bouajina
    • 2
  • Amani Mankaï
    • 1
  • Hela Zeglaoui
    • 2
  • Karim Skandrani
    • 3
  • Ibtissem Ghedira
    • 1
    • 4
  1. 1.Laboratory of ImmunologyFarhat Hached HospitalSousseTunisia
  2. 2.Rheumatology UnitFarhat Hached HospitalSousseTunisia
  3. 3.Gastroenterology UnitFarhat Hached HospitalSousseTunisia
  4. 4.Faculty of PharmacyMonastirTunisia

Personalised recommendations