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Published in: Journal of Medical Case Reports 1/2010

Open Access 01-12-2010 | Case report

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

Authors: Alan M Edwards, Hans Hagberg

Published in: Journal of Medical Case Reports | Issue 1/2010

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Abstract

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 H1 and H2 antihistamines, 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 H1 and H2 antihistamines, 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.
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Literature
1.
go back to reference Castells MC: Mastocytosis: classification, diagnosis, and clinical presentation. Allergy Asthma Proc. 2004, 25 (1): 33-36.PubMed Castells MC: Mastocytosis: classification, diagnosis, and clinical presentation. Allergy Asthma Proc. 2004, 25 (1): 33-36.PubMed
2.
go back to reference Cox JS, Beach JE, Blair AM, Clarke AJ, King J, Lee TB, Loveday DE, Moss GE, Orr TS, Ritchie JT, Sheard P: Disodium cromoglycate (Intal). Adv Drug Res. 1970, 5: 115-196.PubMed Cox JS, Beach JE, Blair AM, Clarke AJ, King J, Lee TB, Loveday DE, Moss GE, Orr TS, Ritchie JT, Sheard P: Disodium cromoglycate (Intal). Adv Drug Res. 1970, 5: 115-196.PubMed
3.
go back to reference Aswania QA, Corlett SA, Chrystyn H: Relative bioavailability of sodium cromoglycate to the lung following inhalation, using urinary excretion. Br J Clin Pharmacol. 1999, 613-618. 10.1046/j.1365-2125.1999.00937.x. 6 Aswania QA, Corlett SA, Chrystyn H: Relative bioavailability of sodium cromoglycate to the lung following inhalation, using urinary excretion. Br J Clin Pharmacol. 1999, 613-618. 10.1046/j.1365-2125.1999.00937.x. 6
4.
go back to reference Walker SR, Evans ME, Richards AJ, Paterson JW: The fate of (14 C) disodium cromoglycate in man. J Pharm Pharmacol. 1972, 24 (7): 525-531.CrossRefPubMed Walker SR, Evans ME, Richards AJ, Paterson JW: The fate of (14 C) disodium cromoglycate in man. J Pharm Pharmacol. 1972, 24 (7): 525-531.CrossRefPubMed
5.
go back to reference Dolovich J, Punthakee ND, MacMillan AB, Osbaldeston GJ: Systemic mastocytosis: control of lifelong diarrhea by ingested disodium cromoglycate. Can Med Assoc J. 1974, 111 (7): 684-685.PubMedPubMedCentral Dolovich J, Punthakee ND, MacMillan AB, Osbaldeston GJ: Systemic mastocytosis: control of lifelong diarrhea by ingested disodium cromoglycate. Can Med Assoc J. 1974, 111 (7): 684-685.PubMedPubMedCentral
6.
go back to reference Soter NA, Austen KF, Wasserman SI: Oral disodium cromoglycate in the treatment of systemic mastocytosis. N Engl J Med. 1979, 301 (9): 465-469.CrossRefPubMed Soter NA, Austen KF, Wasserman SI: Oral disodium cromoglycate in the treatment of systemic mastocytosis. N Engl J Med. 1979, 301 (9): 465-469.CrossRefPubMed
7.
go back to reference Czarnetzki BM, Behrendt H: Urticaria pigmentosa: clinical picture and response to oral disodium cromoglycate. Br J Dermatol. 1981, 105 (5): 563-567. 10.1111/j.1365-2133.1981.tb00800.x.CrossRefPubMed Czarnetzki BM, Behrendt H: Urticaria pigmentosa: clinical picture and response to oral disodium cromoglycate. Br J Dermatol. 1981, 105 (5): 563-567. 10.1111/j.1365-2133.1981.tb00800.x.CrossRefPubMed
8.
go back to reference Frieri M, Alling DW, Metcalfe DD: 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. 1985, 78 (1): 9-14. 10.1016/0002-9343(85)90454-1.CrossRefPubMed Frieri M, Alling DW, Metcalfe DD: 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. 1985, 78 (1): 9-14. 10.1016/0002-9343(85)90454-1.CrossRefPubMed
9.
go back to reference Horan RF, Sheffer AL, Austen KF: Cromolyn sodium in the management of systemic mastocytosis. J Allergy Clin Immunol. 1990, 85 (5): 852-855. 10.1016/0091-6749(90)90067-E.CrossRefPubMed Horan RF, Sheffer AL, Austen KF: Cromolyn sodium in the management of systemic mastocytosis. J Allergy Clin Immunol. 1990, 85 (5): 852-855. 10.1016/0091-6749(90)90067-E.CrossRefPubMed
10.
go back to reference Basomba A, Campos IG, Villalmanzo , Pelaez A: The effect of sodium cromoglycate (SCG) in patients with food allergies. Acta Allergol Suppl. 1977, 13: 95-101. Basomba A, Campos IG, Villalmanzo , Pelaez A: The effect of sodium cromoglycate (SCG) in patients with food allergies. Acta Allergol Suppl. 1977, 13: 95-101.
11.
go back to reference Carini C, Brostoff J: Evidence for circulating IgE complexes in food allergy. Ric Clin Lab. 1987, 17 (4): 309-322.PubMed Carini C, Brostoff J: Evidence for circulating IgE complexes in food allergy. Ric Clin Lab. 1987, 17 (4): 309-322.PubMed
12.
go back to reference Villar SS, Perez-Somarriba J, Costa TS, Winstanley S, Escribano L, Gonzalez B: Interactive Case Report: A 38 year old woman with hypotensive shock at the onset of menstruation. Br Med J. 2009, 338: 654-655. 10.1136/bmj.b868.CrossRef Villar SS, Perez-Somarriba J, Costa TS, Winstanley S, Escribano L, Gonzalez B: Interactive Case Report: A 38 year old woman with hypotensive shock at the onset of menstruation. Br Med J. 2009, 338: 654-655. 10.1136/bmj.b868.CrossRef
13.
go back to reference Andre F, Andre C, Feknous M, Colin L, Cavagna S: Digestive permeability to different-sized molecules and to sodium cromoglycate in food allergy. Allergy Proc. 1991, 12 (5): 293-298. 10.2500/108854191778879142.CrossRefPubMed Andre F, Andre C, Feknous M, Colin L, Cavagna S: Digestive permeability to different-sized molecules and to sodium cromoglycate in food allergy. Allergy Proc. 1991, 12 (5): 293-298. 10.2500/108854191778879142.CrossRefPubMed
14.
go back to reference Auty RM, Brown K, Neale MG, Snashall PD: Respiratory tract deposition of sodium cromoglycate is highly dependent upon technique of inhalation using the Spinhaler. Br J Dis Chest. 1987, 81 (4): 371-380. 10.1016/0007-0971(87)90186-0.CrossRefPubMed Auty RM, Brown K, Neale MG, Snashall PD: Respiratory tract deposition of sodium cromoglycate is highly dependent upon technique of inhalation using the Spinhaler. Br J Dis Chest. 1987, 81 (4): 371-380. 10.1016/0007-0971(87)90186-0.CrossRefPubMed
15.
go back to reference Gerth van Wijk R: From statistical significance to clinical relevance. Clin Experimental Allergy. 2010, 40: 197-199. 10.1111/j.1365-2222.2009.03422.x.CrossRef Gerth van Wijk R: From statistical significance to clinical relevance. Clin Experimental Allergy. 2010, 40: 197-199. 10.1111/j.1365-2222.2009.03422.x.CrossRef
Metadata
Title
Oral and inhaled sodium cromoglicate in the management of systemic mastocytosis: a case report
Authors
Alan M Edwards
Hans Hagberg
Publication date
01-12-2010
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2010
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/1752-1947-4-193

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