Oral and inhaled sodium cromoglicate in the management of systemic mastocytosis: A case report

被引:8
作者
Edwards A.M. [1 ]
Hagberg H. [2 ]
机构
[1] David Hide Asthma and Allergy Research Centre, St Mary's Hospital, Newport
[2] Department of Oncology, University Hospital
关键词
Bone Pain; Mastocytosis; Desloratadine; Systemic Mastocytosis; Allergic Conjunctivitis;
D O I
10.1186/1752-1947-4-193
中图分类号
学科分类号
摘要
Introduction. Mastocytosis is a rare disease consisting of a group of disorders characterized by a pathologic increase in the number of mast cells in one or more organ system. Treatment is symptomatic. Oral sodium cromoglicate (SCG) is the only treatment licensed for the treatment of mastocytosis. In this case we report how in a mastocytosis patient being treated with H1and H2antihistamines, and oral sodium cromoglicate, the addition of inhaled sodium cromoglicate resulted in further improvement. This is the first report of this use of the drug in this disease. Case presentation. The subject is a Caucasian woman aged 40 years. Symptoms of mastocytosis began when she was aged 13 years, but the diagnosis was not made until after her first pregnancy aged 33 years. Symptoms improved with H1and H 2antihistamines, and oral sodium cromoglicate, but it required the addition of inhaled sodium cromoglicate to produce further improvement, specifically in the symptoms of bone pain, fatigue and headache. Doses of oral sodium cromoglicate had to be increased if challenged with a food to which the subject was sensitive. Doses of inhaled sodium cromoglicate had to be increased during the menstrual period. Conclusions. Patients suffering from the rare disease of mastocytosis have symptoms affecting many body systems. Symptoms result from the release of inflammatory mediators from mast cells. Sodium cromoglicate, a drug that reduces the release of mediators from mast cells, is effective in controlling gastrointestinal symptoms, but less effective in those affecting other body systems. In this case report we have shown that the addition of inhaled sodium cromoglicate controls the symptoms of bone pain, fatigue and headache and also that the doses have to be increased during the menstrual period. © 2010 Edwards and Hagberg; licensee BioMed Central Ltd.
引用
收藏
相关论文
共 15 条
  • [1] Castells M.C., Mastocytosis: Classification, diagnosis, and clinical presentation, Allergy Asthma Proc, 25, 1, pp. 33-36, (2004)
  • [2] Cox J.S., Beach J.E., Blair A.M., Clarke A.J., King J., Lee T.B., Loveday D.E., Moss G.E., Orr T.S., Ritchie J.T., Sheard P., Disodium cromoglycate (Intal), Adv Drug Res, 5, pp. 115-196, (1970)
  • [3] Aswania Q.A., Corlett S.A., Chrystyn H., Relative bioavailability of sodium cromoglycate to the lung following inhalation, using urinary excretion, Br J Clin Pharmacol, 6, pp. 613-618, (1999)
  • [4] Walker S.R., Evans M.E., Richards A.J., Paterson J.W., The fate of (14 C) disodium cromoglycate in man, J Pharm Pharmacol, 24, 7, pp. 525-531, (1972)
  • [5] Dolovich J., Punthakee N.D., MacMillan A.B., Osbaldeston G.J., Systemic mastocytosis: Control of lifelong diarrhea by ingested disodium cromoglycate, Can Med Assoc J, 111, 7, pp. 684-685, (1974)
  • [6] Soter N.A., Austen K.F., Wasserman S.I., Oral disodium cromoglycate in the treatment of systemic mastocytosis, N Engl J Med, 301, 9, pp. 465-469, (1979)
  • [7] Czarnetzki B.M., Behrendt H., Urticaria pigmentosa: Clinical picture and response to oral disodium cromoglycate, Br J Dermatol, 105, 5, pp. 563-567, (1981)
  • [8] Frieri M., Alling D.W., Metcalfe D.D., Comparison of the therapeutic efficacy of cromolyn sodium with that of combined chlorpheniramine and cimetidine in systemic mastocytosis. Results of a double-blind clinical trial, Am J Med, 78, 1, pp. 9-14, (1985)
  • [9] Horan R.F., Sheffer A.L., Austen K.F., Cromolyn sodium in the management of systemic mastocytosis, J Allergy Clin Immunol, 85, 5, pp. 852-855, (1990)
  • [10] Basomba A., Campos A., Villalmanzo I.G., Pelaez A., The effect of sodium cromoglycate (SCG) in patients with food allergies, Acta Allergologica, 32, SUPPL. 13, pp. 95-101, (1977)