Case Report: Cerebral venous thrombosis revealing celiac disease

Celiac disease (CD) is an autoimmune enteropathy resulting from intolerance of an individual genetically predisposed to gluten. It has a large clinical polymorphism ranging from a classic digestive clinical presentation due to the malabsorption syndrome to extra-intestinal symptoms. Among the hematologic abnormalities, venous thromboembolic disease (VTE) has been reported, and they are most often located in the abdomen or lower limbs, but the cerebral localization was exceptionally described. We report a case of CD revealed by cerebral thrombophlebitis. A 44-year-old patient with no medical history and no drug intake, presented with hemiplegia followed by a status epilepticus in a context of apyrexia, initially hospitalized in intensive care. Magnetic imaging resonance displayed a cerebral venous thrombosis of the sigmoid sinus requiring anticoagulant treatment, then transferred to our department for the etiological investigation. On questioning, the patient reported chronic diarrhea and weight loss with no other associated symptoms. The examination revealed an underweight patient with pale conjunctiva, improvement of her deficit symptoms, and no other abnormalities. Laboratory tests noted biological signs of malabsorption. The thrombophilia assessment revealed a protein C deficiency with a slight increase in anticardiolipin antibodies and anti-Beta 2 glycoprotein 1 antibodies. Immunological tests noted positives anti-transglutaminase and IgA anti-endomysium antibodies. Duodenal biopsy demonstrated villous atrophy. After ruling out the other causes of VTE, the diagnosis of cerebral venous thrombosis secondary to CD was retained. Early diagnosis and treatment of CD improves the quality-of-life for patients and may spare them various long-term or even fatal complications.

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Cerebral Venous Thrombosis in an Adolescent with Celiac Disease

Scholars Journal of Medical Case Reports ◽

10.36347/sjmcr.2020.v08i04.024 ◽

2020 ◽

Vol 08 (04) ◽

pp. 502-504

Author(s):

Sara Sabiry ◽

Salma Louzi ◽

Zainab Abdulhakeem ◽

Hicham EL Otmani ◽

Mohamed Abdoh Rafai ◽

...

Keyword(s):

Celiac Disease ◽

Venous Thrombosis ◽

Cerebral Venous Thrombosis

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Cerebral Venous Thrombosis and Anticardiolipin Antibodies

Stroke ◽

10.1161/01.str.28.12.2363 ◽

1997 ◽

Vol 28 (12) ◽

pp. 2363-2369 ◽

Cited By ~ 68

Author(s):

Juan R. Carhuapoma ◽

Panayiotis Mitsias ◽

Steven R. Levine

Keyword(s):

Venous Thrombosis ◽

Cerebral Venous Thrombosis ◽

Anticardiolipin Antibodies

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Recurrent Cerebral Venous Thrombosis Treated with Direct Oral Anticoagulants in a Japanese Man with Hereditary Protein C Deficiency

Journal of Stroke and Cerebrovascular Diseases ◽

10.1016/j.jstrokecerebrovasdis.2020.105320 ◽

2021 ◽

Vol 30 (1) ◽

pp. 105320

Author(s):

Kazuyuki Saito ◽

Kazuyuki Ishii ◽

Konosuke Furuta ◽

Masaki Kobayashi ◽

Yoshiaki Wada ◽

...

Keyword(s):

Venous Thrombosis ◽

Cerebral Venous Thrombosis ◽

Protein C ◽

Oral Anticoagulants ◽

Direct Oral Anticoagulants ◽

Protein C Deficiency

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Cerebral venous thrombosis as a rare thromboembolic complication of celiac disease: a case report

European Heart Journal - Case Reports ◽

10.1093/ehjcr/ytaa200 ◽

2020 ◽

Vol 4 (5) ◽

pp. 1-5

Author(s):

Maha Bouziane ◽

Salim Arous ◽

Rachida Habbal

Keyword(s):

Celiac Disease ◽

Venous Thrombosis ◽

Cerebral Venous Thrombosis ◽

Sinus Thrombosis ◽

Anticoagulation Therapy ◽

Thromboembolic Complication ◽

Favourable Outcome ◽

Complete Regression ◽

Thromboembolic Events ◽

Cerebral Magnetic Resonance Imaging

Abstract Background Celiac disease is a digestive inflammatory syndrome with several complications. It is associated with coagulation and platelets abnormalities leading to thromboembolic events. Cerebral venous thrombosis is an exceptional localization of thrombosis in celiac disease and could be life-threatening. Case summary A 17-year-old female patient with history of celiac disease and not following a gluten-free diet, checked in to the emergency department for a sudden, 2-week-old, and deteriorating, onset of intense headache and muscle weakness. The cerebral computed tomography-scan showed bilateral fronto-parietal hypodensity with micro-bleeds. We investigated using a cerebral magnetic resonance imaging that revealed superior longitudinal sinus thrombosis and right transverse and sigmoid sinuses thrombosis, along with right haematoma and ischaemic areas. The patient was prescribed anticoagulation therapy. Follow-ups over a 2-year period confirmed a favourable outcome and a complete regression of symptoms. Discussion Evolution of celiac disease could be associated with several complications. Eighty-five percent of patients is potentially exposed to thromboembolic events due to the hypercoagulability state of the disease and different coagulation and fibrinolysis abnormalities (e.g. hyperhomocysteinaemia, protein C and S deficiencies, vitamin K and B deficiencies). Cerebral venous thrombosis is a rare thromboembolic localization. Anticoagulation is efficient in most cases though endovascular treatment might be required.

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Recurrent cerebral venous thrombosis revealing celiac disease: an exceptional case report

Acta Neurologica Belgica ◽

10.1007/s13760-016-0698-y ◽

2016 ◽

Vol 117 (1) ◽

pp. 341-343 ◽

Cited By ~ 5

Author(s):

Rahma Beyrouti ◽

Malek Mansour ◽

Amel Kacem ◽

Hager Derbali ◽

Ridha Mrissa

Keyword(s):

Celiac Disease ◽

Case Report ◽

Venous Thrombosis ◽

Cerebral Venous Thrombosis ◽

Exceptional Case

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Cerebral haemorrhagic infarction in young patients with hereditary protein C deficiency: evidence for "spontaneous" cerebral venous thrombosis.

BMJ ◽

10.1136/bmj.290.6465.350 ◽

1985 ◽

Vol 290 (6465) ◽

pp. 350-352 ◽

Cited By ~ 46

Author(s):

A R Wintzen ◽

A W Broekmans ◽

R M Bertina ◽

E Briet ◽

P E Briet ◽

...

Keyword(s):

Venous Thrombosis ◽

Cerebral Venous Thrombosis ◽

Protein C ◽

Young Patients ◽

Protein C Deficiency ◽

Haemorrhagic Infarction

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Arterial events, venous thromboembolism, thrombocytopenia, and bleeding after vaccination with Oxford-AstraZeneca ChAdOx1-S in Denmark and Norway: population based cohort study

BMJ ◽

10.1136/bmj.n1114 ◽

2021 ◽

pp. n1114 ◽

Cited By ~ 1

Author(s):

Anton Pottegård ◽

Lars Christian Lund ◽

Øystein Karlstad ◽

Jesper Dahl ◽

Morten Andersen ◽

...

Keyword(s):

Cohort Study ◽

Venous Thromboembolism ◽

Venous Thrombosis ◽

Cerebral Venous Thrombosis ◽

Population Based ◽

Thromboembolic Events ◽

Coagulation Disorders ◽

Venous Thromboembolic Events ◽

Venous Thromboembolic ◽

General Populations

Abstract Objective To assess rates of cardiovascular and haemostatic events in the first 28 days after vaccination with the Oxford-AstraZeneca vaccine ChAdOx1-S in Denmark and Norway and to compare them with rates observed in the general populations. Design Population based cohort study. Setting Nationwide healthcare registers in Denmark and Norway. Participants All people aged 18-65 years who received a first vaccination with ChAdOx1-S from 9 February 2021 to 11 March 2021. The general populations of Denmark (2016-18) and Norway (2018-19) served as comparator cohorts. Main outcome measures Observed 28 day rates of hospital contacts for incident arterial events, venous thromboembolism, thrombocytopenia/coagulation disorders, and bleeding among vaccinated people compared with expected rates, based on national age and sex specific background rates from the general populations of the two countries. Results The vaccinated cohorts comprised 148 792 people in Denmark (median age 45 years, 80% women) and 132 472 in Norway (median age 44 years, 78% women), who received their first dose of ChAdOx1-S. Among 281 264 people who received ChAdOx1-S, the standardised morbidity ratio for arterial events was 0.97 (95% confidence interval 0.77 to 1.20). 59 venous thromboembolic events were observed in the vaccinated cohort compared with 30 expected based on the incidence rates in the general population, corresponding to a standardised morbidity ratio of 1.97 (1.50 to 2.54) and 11 (5.6 to 17.0) excess events per 100 000 vaccinations. A higher than expected rate of cerebral venous thrombosis was observed: standardised morbidity ratio 20.25 (8.14 to 41.73); an excess of 2.5 (0.9 to 5.2) events per 100 000 vaccinations. The standardised morbidity ratio for any thrombocytopenia/coagulation disorders was 1.52 (0.97 to 2.25) and for any bleeding was 1.23 (0.97 to 1.55). 15 deaths were observed in the vaccine cohort compared with 44 expected. Conclusions Among recipients of ChAdOx1-S, increased rates of venous thromboembolic events, including cerebral venous thrombosis, were observed. For the remaining safety outcomes, results were largely reassuring, with slightly higher rates of thrombocytopenia/coagulation disorders and bleeding, which could be influenced by increased surveillance of vaccine recipients. The absolute risks of venous thromboembolic events were, however, small, and the findings should be interpreted in the light of the proven beneficial effects of the vaccine, the context of the given country, and the limitations to the generalisability of the study findings.

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