Contributions
Abstract: P11
Type: Poster presentation
Presentation during EHA Scientific Conference on Bleeding Disorders:
On Friday, September 16, 2016 from 14:00 - 15:30
Location: Cristal + Coral
Background
Fingolimod is an oral sphingosine-1-phosphate–receptor modulator which causes lymphocyte sequestration in lymph nodes. It was approved for relapsing Multiple Sclerosis (MS) following evidence it reduced relapse rates by 50%1,2. The Therapeutic Goods Administration (TGA) of Australia as of September 2015 was aware of only one case where fingolimod preceded ITP by five weeks.
Aims
To report three cases of ITP associated with fingolimod.
Methods
We retrospectively reviewed three cases of fingolimod associated ITP who presented to Monash Health between 2013 and 2015.
Results
Cases are described in Table 1. None were on any medications known to cause ITP and routine investigations were non-contributory. All cases were treated with immunosuppression. Case 1 successfully weaned prednisolone after fingolimod cessation whilst case 2 had a slower wean whilst continuing fingolimod therapy. Case 3 had more refractory ITP and re-exposure to fingolimod worsened thrombocytopenia.
Possible mechanisms of the potential association between fingolimod and ITP remain unclear. One possible theory is immune dysregulation given fingolimod has been associated with autoimmune haemolytic anaemia3 and haemophagocytic syndrome4,5. Another could be that it merely highlights autoimmune clustering.
| Case 1 | Case 2 | Case 3 | |
| Year at ITP Diagnosis | 2014 | 2013 | 2015 |
| Age (years) | 22 | 51 | 59 |
| MS Duration (years) | 3 | 2 | 10 |
| Previous MS Therapy | Beta interferon | Beta interferon | Methylprednisolone, Dimethyl fumarate |
| Other Autoimmune Conditions | - | Rheumatoid Arthritis Graves' Disease | - |
| Duration of Fingolimod prior to ITP | 12 months | 2 months | 19 months |
| Fingolimod post ITP | Continued | Continued | Discontinued |
| ITP Treatment | Prednisolone | Prednisolone, IVIG, Azathioprine, Hydroxychloroquine | Prednisolone, IVIG, Azathioprine, Hydroxychloroquine, Elthrombopag, Romiplostim |
In conclusion, our cases highlight that clinicians should be aware of the possible association between ITP and fingolimod although the mechanism for this remains unclear.
References
1. Cohen JA et al, N Engl J Med. 2010;362(5)
2. Kappos et al, The N Engl J Med. 2010 362(5)
3. Lysandropoulos et al, Mult Scler 2013 19(11)
4. Abreu P et al, Neurology. 2014 82(10 Supplement).
5. Ikumi K et al Neurol Neuroimmunol Neuroinflamm. 2016 3(4)
Abstract: P11
Type: Poster presentation
Presentation during EHA Scientific Conference on Bleeding Disorders:
On Friday, September 16, 2016 from 14:00 - 15:30
Location: Cristal + Coral
Background
Fingolimod is an oral sphingosine-1-phosphate–receptor modulator which causes lymphocyte sequestration in lymph nodes. It was approved for relapsing Multiple Sclerosis (MS) following evidence it reduced relapse rates by 50%1,2. The Therapeutic Goods Administration (TGA) of Australia as of September 2015 was aware of only one case where fingolimod preceded ITP by five weeks.
Aims
To report three cases of ITP associated with fingolimod.
Methods
We retrospectively reviewed three cases of fingolimod associated ITP who presented to Monash Health between 2013 and 2015.
Results
Cases are described in Table 1. None were on any medications known to cause ITP and routine investigations were non-contributory. All cases were treated with immunosuppression. Case 1 successfully weaned prednisolone after fingolimod cessation whilst case 2 had a slower wean whilst continuing fingolimod therapy. Case 3 had more refractory ITP and re-exposure to fingolimod worsened thrombocytopenia.
Possible mechanisms of the potential association between fingolimod and ITP remain unclear. One possible theory is immune dysregulation given fingolimod has been associated with autoimmune haemolytic anaemia3 and haemophagocytic syndrome4,5. Another could be that it merely highlights autoimmune clustering.
| Case 1 | Case 2 | Case 3 | |
| Year at ITP Diagnosis | 2014 | 2013 | 2015 |
| Age (years) | 22 | 51 | 59 |
| MS Duration (years) | 3 | 2 | 10 |
| Previous MS Therapy | Beta interferon | Beta interferon | Methylprednisolone, Dimethyl fumarate |
| Other Autoimmune Conditions | - | Rheumatoid Arthritis Graves' Disease | - |
| Duration of Fingolimod prior to ITP | 12 months | 2 months | 19 months |
| Fingolimod post ITP | Continued | Continued | Discontinued |
| ITP Treatment | Prednisolone | Prednisolone, IVIG, Azathioprine, Hydroxychloroquine | Prednisolone, IVIG, Azathioprine, Hydroxychloroquine, Elthrombopag, Romiplostim |
In conclusion, our cases highlight that clinicians should be aware of the possible association between ITP and fingolimod although the mechanism for this remains unclear.
References
1. Cohen JA et al, N Engl J Med. 2010;362(5)
2. Kappos et al, The N Engl J Med. 2010 362(5)
3. Lysandropoulos et al, Mult Scler 2013 19(11)
4. Abreu P et al, Neurology. 2014 82(10 Supplement).
5. Ikumi K et al Neurol Neuroimmunol Neuroinflamm. 2016 3(4)