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CASE REPORT
Ahead of print publication  

Unusual presentation of cerebral venous sinus thrombosis in ulcerative colitis


 Department Internal Medicine, AFMC, Pune, Maharashtra, India

Date of Submission21-Apr-2020
Date of Decision25-May-2020
Date of Acceptance11-Jun-2020
Date of Web Publication21-Sep-2020

Correspondence Address:
Adwait Sodani,
Department of Internal Medicine, Armed Forces Medical College, Pune - 411 040, Maharashtra
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jmms.jmms_41_20

  Abstract 


A 21-year-old young male presented with a relapse of Idiopathic Ulcerative Colitis (IUC) and developed recurrent transient weakness of left upper limb with complete recovery and headache. He also had blurring of vision and pre-syncope. The evaluation revealed Cerebral Venous Thrombosis (CVT). The attacks seized to occur after prompt anticoagulation was exhibited. The purpose of this report is to emphasize that CVT in IUC is sparsely experienced a prothrombotic complication and such CVT presenting as unilateral recurrent transient paresis or some focal deficit mimicking a Transient Ischemic Attack (TIA) / seizure is further rare albeit possible.

Keywords: Cerebral venous thrombosis, idiopathic ulcerative colitis, recurrent monoparesis, seizure



How to cite this URL:
Manrai M, Mohakuda S S, Sodani A. Unusual presentation of cerebral venous sinus thrombosis in ulcerative colitis. J Mar Med Soc [Epub ahead of print] [cited 2020 Nov 24]. Available from: https://www.marinemedicalsociety.in/preprintarticle.asp?id=295638




  Introduction Top


Venous thrombosis may occur in inflammatory bowel disease (IBD), however of the cerebral circulation is lower on cards with only 1.6% of cerebral venous thrombosis (CVT) attributable to IBD.[1] Further, CVT is more common in females but our case is a male; an observation consistent with a CVT due to IBD.[2] It is headache or seizure that brings this diagnosis to attention while focal deficits, like in our case, are rare. More so, the pattern of recurrent transient deficits was hitherto sparsely heard off.


  Case Report Top


A 21-year-old male, with no addictions, was a known case of idiopathic ulcerative colitis (IUC) - Pancolitis E4 disease (onset Jun 2017), who initially presented with multiple episodes of painful, bloody diarrhea with mucus and constitutional symptoms and was diagnosed as a case of IUC after a colonoscopy [Figure 1] which revealed multiple superficial ulcerations and edema. Colonic biopsy revealed the features of IUC. His remission was induced with prednisolone, and infliximab and maintained with mesalamine, and azathioprine for about a year until now when he presented with fever, diffuse pain abdomen and an increased frequency of stools (6–8 times), mixed with blood and mucus with associated anorexia, and weight loss. There was no relevant past or family history.
Figure 1: Colonoscopy showing extensive ulceration of the mucosa

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On examination, he was alert, oriented, and afebrile with tachycardia (100/min regular), supine blood pressure of 118/76 mmHg, and pallor. There was no pedal edema, lymphadenopathy, clubbing, icterus, or cyanosis. The abdomen was soft but tender with diffuse guarding.

Laboratory evaluation revealed hemoglobin of 7.2 gm%; Total Leucocyte Count (TLC) – 13,700/mL; platelet – 7.7 lac/mL, ESR – 26 mm, PBS – microcytic hypochromic anemia, urea/creatinine – 12/0.6 mg/dl; Na/K-144/4.9 mEq/L, bilirubin total/direct – 0.3/0.1 mg%, total protein/albumin/globulin – 5.9/2.5/3.4 g/dl; SGOT/PT/ALP – 117/161/76 IU/L, stool routine and microscopy – numerous pus cells, red blood cells +, no ova cyst or fat globules were noted, viral markers (anti HCV/HBsAg/HIV) were negative, PT/INR/aPTT – 15/1.2/38 s.

He was managed as a case of acute exacerbation (Truelove and Witts score – severe) of IUC with intravenous steroids (followed by tapering oral doses), empirical intravenous antibiotics (ciprofloxacin and metronidazole), and provided blood component support (PRBC) for anemia. The patient responded to treatment (stool frequency 1–2/day) without blood, mucus, or pain and was put on oral mesalamine and azathioprine.

Later during convalescence, still in the hospital, he started to suffer with recurrent transient sudden onset episodic weakness of left upper extremity (LUE) in the form that he was unable to tie his pajamas or lift LUE above his head; which culminated with complete spontaneous recovery within 10 min each time. During these episodes, he remained alert, oriented, afebrile, and with stable vitals. There were no abnormal movements of LUE which on examination had hypotonia with distal more than proximal weakness. Deep-tendon reflexes and sensations in LUE remained normal. All other extremities were clinically normal.

Repeat laboratory evaluation suggested persisting anemia (9 gm%), but routine biochemistry was unremarkable. Noncontrast computed tomography head [Figure 2] done after the first episode showed hyperdensities suggestive of Cerebral Venous Thrombosis (CVT) involving superior sagittal, left transverse, and left sigmoid sinuses and the left jugular bulb. Later, an magnetic resonance imaging with magnetic resonance venography [Figure 3] confirmed the same. Pro-coagulation workup revealed the presence of CD55/59 at 95%, LAC, APLA, and ACLA were negative while ANA (ELIZA) was 0.5 units.
Figure 2: Noncontrast computed tomography head shows hyperdense lesions suggestive of dural venous sinus thrombosis (arrows: Part 1: Superior sagittal sinus and Part 2 transverse and sigmoid sinus)

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Figure 3: Magnetic resonance imaging venogram brain confirming multiple dural venous sinus thrombosis (arrows). No parenchymal lesions noted

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He was managed with low-molecular-weight heparin and responded with no residual motor weakness. He was discharged on warfarin (INR optimized) with advice for continued OPD follow-up. On follow-up, there was no recurrence noted. The required consent was taken from the patient for onward publication of his case.


  Discussion Top


CVT, a rare but well-recognized extraintestinal complication of IBD, remains a challenging diagnosis because of its myriad presentations.[3] The most frequently involved vessels are the transverse sinus followed by superior sagittal sinus.

Such neurovascular complications occur in 1.3%–6.4% of IBD (UC more than Crohn's disease), and they are multifactorial. Few mechanisms are the deficiency of folate, Vitamin B12, and Vitamin B6 (causing hyperhomocysteinemia), endothelial dysfunction, and activation of platelets. However, they have no correlation with disease activity or with the duration of the disease or with the dose of steroids used in management.[4],[5] These events are common especially during acute exacerbations or relapse because of the associated hypercoagulable state that ensues. Eventually, this state may reverse to normal between exacerbation.[6],[7]

CVT in IBD commonly present as headaches (70%–90%), seizures, decreased consciousness, visual changes, or focal neurological deficit (FND – which are relatively uncommon). FND when present, mostly persists.[5] However, it is was quite interesting to find herein that the attacks of monoparesis completely reversed without any treatment within a few minutes; and also that these were recurrent.

Interestingly though, FND may precede the intestinal symptoms by years.[8]

FND in CVT may be motor (most common), sensory, cranial nerve deficits, or aphasias, cortical blindness, etc., with noninflammatory CVTs presenting with such deficits, unlike the infectious CVTs which show up as a cavernous sinus thrombosis. These FNDs are mostly, secondary to large infarcts in the proximal rolandic region near the midline. However, no such lesion was visualized in our case. Such motor deficits could be acute like an arterial stroke or sub-acute like a space-occupying lesion or may very rarely mimic a TIA. Hemiparesis is associated with the poor prognosis in such cases.[9]

Although uncommon, such episodes could well be focal seizures (as they follow suit with the expected subtype - simple partial seizure), yet GTCS as a fact remains more expected when it comes to seizures in this subset of the population.[9]


  Conclusion Top


In conclusion, it is highlighted that CVT, especially in IUC, is a sparsely experienced prothrombotic complication and such a CVT presenting as unilateral recurrent transient monoparesis mimicking a TIA/as recurrent focal onset motor onset aware seizure is still relatively uncommon a manifestation.

Acknowledgment

The authors would like to thank Dr. Priyanka, Dr. Aparna and Dr. Ajoy Sodani.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Conners LM, Ahad R, Janda PH, Mudasir Z. Cerebral Venous Sinus Thrombosis in a Patient with Ulcerative Colitis Flare. Case Rep Neurol Med 2018;2018:1-7.  Back to cited text no. 1
    
2.
Casella G, Tontini GE, Bassotti G, Pastorelli L, Villanacci V, Spina L, et al. Neurological disorders and inflammatory bowel diseases. World J Gastroenterol 2014;20:8764-82.  Back to cited text no. 2
    
3.
Suda S, Katsura K, Okubo S, Abe A, Suzuki K, Suzuki M, et al. Successful treatment of cerebral venous thrombosis associated with ulcerative colitis. J Stroke Cerebrovasc Dis 2013;22:e684-6.  Back to cited text no. 3
    
4.
Cojocaru IM, Cojocaru M, Sapira V, Ionescu A, Tacu N. Cerebrovascular complications in patients with inflammatory bowel disease. Rom J Intern Med 2014;52:39-44.  Back to cited text no. 4
    
5.
Katsanos AH, Katsanos KH, Kosmidou M, Giannopoulos S, Kyritsis AP, Tsianos EV. Cerebral sinus venous thrombosis in inflammatory bowel diseases. QJM 2013;106:401-13.  Back to cited text no. 5
    
6.
Taous A, Berri MA, Lamsiah T, Zainoun B, Ziadi T, Rouimi A. Cerebral venous thrombosis revealing an ulcerative colitis. Pan Afr Med J 2016;23:120.  Back to cited text no. 6
    
7.
Menon B, Goyal R, Nihal L, Reddy R. Cerebral venous thrombosis in ulcerative colitis. J Neurosci Rural Pract 2013;4:84-6.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Nudelman RJ, Rosen DG, Rouah E, Verstovsek G. Cerebral sinus thrombosis: a fatal neurological complication of ulcerative colitis, Patholog Res Int 2010;2010:132754.  Back to cited text no. 8
    
9.
Luo Y, Tian X, Wang X. Diagnosis and treatment of cerebral venous thrombosis: A review. Front Aging Neurosci 2018;10:2.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

 
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