CASE REPORT Journal of the College of Physicians and Surgeons Pakistan 2020, Vol. 30(07): 762-764 762 Neurological Sequelae in Youngest Adult Behcet’s Disease Patient on Infliximab Mamoona Sultan 1 , Adeena Khan 2 , Syed Shahid Habib 3 and Muhammad Hayat 4 1 Department of Medicine, King Abdul Aziz University Hospital, King Saud University Medical City, Riyadh, Kingdom of Saudi Arabia 2 Department of Radiology, King Abdul Aziz University Hospital, King Saud University Medical City, Riyadh, Kingdom of Saudi Arabia 3 Department of Physiology, King Abdul Aziz University Hospital, King Saud University Medical City, Riyadh, Kingdom of Saudi Arabia 4 ICU, Sanad Hospital, Riyadh, Saudi Arabia ABSTRACT Neuro-Behcet ’ s disease is a rare neurological complication of Behcet ’ s disease presenting between second and fourth decades of life. The etiology of Behcet ’ s disease is still unknown but genetic predisposition has been described to play a role. The disease is reported more in males than females. Neurological involvement could cause brain parenchymal, vascular or meningeal changes with other distinct systemic symptoms, which may include orogenital ulcers, uveitis and skin lesions. Parenchymal neuro-Behcet ’ s disease has a poor prognosis. MRI aids in diagnosing and differentiating it from other entities. Treatment includes anti-inflammatory drugs and immunotherapeutics. Our case is the youngest adult reported so far and is an unusual presentation of neuro-Behcet ’ s disease, as the patient was already in remission and on infliximab infusion. Key Words: Neuro-Behcet’s, MRI, Infliximab. How to cite this article: Sultan M, Khan A, Habib SS, Hayat M. Neurological Sequelae in Youngest Adult Behcet’s Disease Patient on Infliximab. J Coll Physicians Surg Pak 2020; 30(07):762-764. INTRODUCTION Behcet’s disease is a rare multisystem multivessel autoimmune relapsing remitting disorder of unknown etiology, reported most frequently in Turkey with prevalence of 20 - 602/ 100,000. 1,2 Neuro- Behcet’s is also rare but serious complication of Behcet's disease seen in 5 to 50% of patients. 3 Behcet ’ s, a Turkish dermatologist, described its triad of recurrent symptoms in 1937 for the first time, i.e., oral ulcers, genital ulcer and uveitis. 2,4 The term “Neuro-Be- hçet’s syndrome” was first presented by Cavara and D’Ermo in 1954. 5 Neurological complications are commonly due to brain parenchymal involvement. 2 After the first reported case by Knapp in 1941, 6 many cases have been reported in the past; but our patient is the youngest adult reported so far in literature, who developed Neuro-Behcet’s symp- toms while on infliximab infusion. CASEREPORT An18-yearmalewasdiagnosed8yearsagowithBehcet’sdisease due to presentation with recurrent oral and genital ulcers. Correspondence to: Dr. Mamoona Sultan, Department of Medicine, King Abdul Aziz University Hospital, King Saud University Medical City, Riyadh, Kingdom of Saudi Arabia E-mail: mamoonasultankauh@gmail.com ..................................................... Received: May 03, 2019; Revised: September 03, 2019; Accepted: September 12, 2019 DOI: https://doi.org/10.29271/jcpsp.2020.07.762 Patient was followed up in King Saud University Medical City for Behcet’s uveitis and was on infliximab infusion for the past 6 years after failed response of uveitis to corticosteroids and mycophenolate mofetil as an initial immunosuppressant therapy. He presented in ophthalmology emergency with severe headache and double vision and was referred to neurology. On 3 rd day of admission, he developed right-sided hemiparesis. Examination findings were ptosis of left eye with limited adduc- tion and elevation, sluggish pupil response and left-sided homonymous hemianopia. Power was reduced to 4/5 in right upper and lower limbs. Blood investigations were grossly normal but cerebrospinal fluid (CSF) was positive for inflammatory changes, i.e. protein level of 0.930 g/L; white blood cells, 50/mm 3 ; polymorphonu- clear cells, 15%; lymphocytes, 85%; and glucose normal. CSF culture and herpes simplex virus 1 and 2 DNA were negative. Nerve conduction studies showed no abnormality. Contrast enhanced MRI brain showed diffuse hyperintense T2/FLAIR signals involving the midbrain including cerebral peduncles,pons,bilateralthalami,middlecerebellarpeduncles and bilateral internal capsules (Figure 1). Internal capsule and thalamiinvolvementwereexplainingrighthemiparesisandleft homonymous hemianopia, while pons involvement was attributing to the third nerve palsy. MR angiogram was normal. Based on the clinical symptoms, CSF and radiological findings, clinical diagnosis of parenchymal Neuro-Behcet’s was made.