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LETTER TO EDITOR
Year : 2004  |  Volume : 52  |  Issue : 2  |  Page : 281-282

Trigger autoimmunity -Development of multiple plexopathy in a patient with chronic idiopathic thrombocytopenic purpura


Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry, India

Correspondence Address:
Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry, India



How to cite this article:
Ghotekar L H, Dutta T K, Narayanan S, Basu D, Dambal A, Vivekanandan. Trigger autoimmunity -Development of multiple plexopathy in a patient with chronic idiopathic thrombocytopenic purpura. Neurol India 2004;52:281-2


How to cite this URL:
Ghotekar L H, Dutta T K, Narayanan S, Basu D, Dambal A, Vivekanandan. Trigger autoimmunity -Development of multiple plexopathy in a patient with chronic idiopathic thrombocytopenic purpura. Neurol India [serial online] 2004 [cited 2021 May 6];52:281-2. Available from: https://www.neurologyindia.com/text.asp?2004/52/2/281/11080


Sir,
A case of chronic idiopathic thrombocytopenic purpura (ITP) who developed plexopathy, diabetes mellitus and transient disseminated intravascular coagulation (DIC) after splenectomy during hospitalization for the treatment of ITP is presented.
A 42-year-old man diagnosed to be having chronic ITP was admitted for elective splenectomy, for a steroid-responsive but dependent status. His diagnosis was reconfirmed during the preoperative period. He underwent splenectomy. On the third postoperative day, his blood sugar was 348 mg/dl with the presence of urinary ketones, and arterial blood gas (ABG) revealed metabolic acidosis. He was treated with plain porcine insulin, intravenous fluid and electrolytes. His ketoacidosis was controlled with a total of 40 U of plain insulin. During the postoperative period his platelet counts remained at 20-24000/cumm, while peripheral smear revealed fragmented RBCs and thrombocytopenia. Paracoagulation test for fibrinogen degradation product (FDP) was positive with prolonged prothrombin time (test 21 min; control 13 min). DIC was diagnosed for which he received fresh frozen plasma (FFP) and platelet packs. Multiple blood cultures, urine cultures, cultures from the site of surgery were negative. On the fifth postoperative day he complained of weakness and numbness of the right upper limb. Clinical examination revealed lower motor neuron weakness and wasting of the following muscles: in the upper limb on the right side serratus anterior, pectoralis major, supraspinatus, infraspinatus, latissimus dorsi and teres major while on the left side, muscles of the thenar and hypothenar group, dorsal and palmar interossei, and the lumbricals were involved. He had a loss of all sensory modality over the right forearm, hand, lower 1/3rd of the right arm and the ulnar border of the left hand and forearm. Deep tender reflexes were lost in the right upper limb. Plain radiography, CT thorax and magnetic imaging resonance (MRI) of the spine were normal. CSF study was normal. Danazol, prednisolone, insulin and physiotherapy were prescribed. At discharge his platelet count was 1,20,000/cumm and perception of sensations and muscle power in both upper limbs was improving. At follow-up after one and a half months, the power also improved, but he required prednisolone and danazol for maintaining a good platelet count.
“Kaleidoscopic autoimmunity” has been reported with various diseases.[1],[2],[3],[4],[5],[6] Our patient developed transient DIC, diabetes mellitus and multiple plexopathy following splenectomy. Multiple plexopathy has a variable presentation. One of the forms is neuralgic amyotrophy (NA) or acute brachial neuritis (ABN) which usually presents as severe pain in the shoulder followed by weakness of shoulder girdle muscles. Moore et al reported a case of lumbosacral plexopathy in a woman with CREST syndrome and vasculitis.[7] The most common mechanism is thought to be viral etiology or immune-mediated. Blood lymphocytes are known to get sensitized to branchial plexus nerves in patients with neuralgic amyotrophy.[8] It is usually unilateral with rare bilateral asymmetrical findings. Sensory symptoms are rare. Sometimes patchy sensory loss may be present. Electrophysiological studies might demonstrate sub-clinical involvement in asymptomatic limbs in up to 25% of patients and very rarely there is a mild lymphocytic pleocytosis or a rise in protein in the CSF.[9] In our patient there was bilateral asymmetrical involvement which is a rare presentation along with weakness without any pain. Our patient also had right common peroneal nerve involvement demonstrated by electrophysiological studies. Prognosis of NA is good with full recovery of strength in 90% of the patients by three years.[10] Plexopathy in our patient was probably immune-mediated. The cause for DIC in our patient was most probably immune-mediated though surgical trauma or diabetic ketoacidosis with severe dehydration contributing to DIC could not be ruled out. 

 » References Top

1.Shoenfeld Y. Kaleidoscopic autoimmunity. Harefuah 1998;135:262-4.  Back to cited text no. 1  [PUBMED]  
2.Sheehan NJ, Stanton-King K. Polyautoimmunity in a young woman. Br Jr Rheumatol 1993;32:254-6.  Back to cited text no. 2  [PUBMED]  
3.Jordan JM, Manning M, Allen NB. Multiple unusual manifestations of Wegener's granulomatosis: Breast mass, microangiopathic hemolytic anemia, consumptive coagulopathy and low erythrocyte sedimentation rate. Arthritis & Rheumatism 1986;29:1527-31.  Back to cited text no. 3  [PUBMED]  
4.Branger B, Zabadani B, Vecina F, Balducchi JP, Fourcade J. Multiple autoimmune syndrome: succession of bullous pemphigoid, endocapillary proliferative glomerulonephritis, Guillain-Barre syndrome and idiopathic thrombocytopenic purpura: Cascade-apheresis or "the panacea"?. Annales de Medecine Interne 1994;145:382-3.  Back to cited text no. 4    
5.Bachmeyer C, Monge M, Dhote R, Sanguina M, Aractingi S, Mougeot-Martin M. Eosinophilic fasciitis following idiopathic thrombocytopenic purpura, autoimmune hemolytic anemia and Hashimoto's disease. Dermatology 1999;199:282.  Back to cited text no. 5    
6.Oliveros-Cid A, Martinez-Vila E, Castillo J. Revista de Neurologia 1997;25:S294-308.  Back to cited text no. 6    
7.Moore ME, Burke JM, Hartman JH, Korenzwitt E. Lumbosacral plexopathy in a woman with CREST syndrome and vasculitis. Arthritis and Rheumatism 1989;32:661-3.  Back to cited text no. 7    
8.Sierra A, Prat J, Bas J, Romeu A, Montero J, Matos JA, et al. Blood lymphocytes are sensitized to branchial plexus nerves in patients with neuralgic amyotrophy. Acta Neurologica Scandinavica 1991;83:183-6.  Back to cited text no. 8    
9.Tsairis P, Dyck, PJ, Mulder DW. Natural history of brachial plexus neuropathy. Arch Neurol 1972;27:109.  Back to cited text no. 9    
10.Schott GD. A chronic and painless form of idiopathic brachial plexus neuropathy. J Neurol Neurosurg Psychiat 1983;46:555.  Back to cited text no. 10    

 

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