Case report long segment myelitis secondary to neuro melioidosis.

BACKGROUND: Neuro-melioidosis, comprising 4% of all cases of melioidosis carries a risk of high morbidity and mortality. We describe two Sri Lankan patients presenting with long segment myelitis secondary to melioidosis. CASE PRESENTATION: Case 1: 47-year-old male presented with right side hemiparesis which progressed rapidly to quadriparesis. Initial cerebro spinal fluid (CSF) analysis revealed protein 76 mg/dl and glucose 72 mg/dl but without a cellular reaction. MRI spine revealed long segment myelitis with contrast enhancement. The patient was treated with intravenous methyl prednisolone pulses (IV MPP) and plasma exchanges(PLEX) on suspicion of an immune mediated myelitis but without success. A repeat MRI revealed high signal changes in the brain stem and along the entire spinal cord with contrast enhancement. MRI brain after treatment with MPP/PLEX showed enhancing hyper intensities along the corticospinal tracts. The repeat CSF revealed protein 1187 mg/dl, glucose 78 mg/dl, lymphocytes 1600/mm3 and neutrophils 10,200/mm3. CSF culture has become positive for Burkholderia pseudomallei. Serum melioidosis antibody titre was 1: 320. He was started on IV meropenem with oral cotrimoxazole for 12 weeks followed by oral co trimoxazole. But he had poor clinical recovery. Case 2: 47-year-old female presented with bilateral lower limb weakness for 1-week duration. On examination, she had flaccid paraparesis with a sensory level at T11. Inflammatory markers were elevated. CSF analysis revealed protein 50 mg/dl with lymphocytes 172/mm3. MRI pan spine revealed a long segment myelitis. Serum melioidosis antibody titre was 1: 640. She was treated with IV meropenem for 8 weeks followed by oral co-trimoxazole with an excellent clinical and radiological response. CONCLUSION: Numerous neurological manifestations have been described with melioidosis, however long segment myelitis with a positive CSF culture is not yet reported. These cases signify the importance of considering melioidosis as a differential in patients with long segment myelitis especially in endemic areas.

Location and Diameter of Intracranial Meningioma as Predictors of Peritumoral Brain Oedema and Mass Effect.

INTRODUCTION: The presence of peritumoral oedema or mass effect with intracranial meningiomas is associated with poor clinical outcomes. This study aimed to investigate magnetic resonance (MR) morphologic features of meningioma, which can predict peritumoral oedema and mass effect. METHODS: Data of 100 consecutive patients diagnosed with meningioma on MRI brain at the neurosurgical MRI unit, National Hospital of Sri Lanka, reported by a Consultant Radiologist were analysed in a retrospective study. Binary logistic regression models were fitted to identify predictors of perilesional oedema and mass effect. RESULTS: Female-to-male ratio was 5.8:1. Patients were aged 18-80 years. Majority (n = 78) were in supratentorial compartment with 16 at parasagittal location. Cerebellopontine angle was the commonest infratentorial site (n = 9). Size of meningiomas ranged from 1.1 to 9.1 cm (largest dimension). Mass effect (n = 68), perilesional oedema (n = 37), and midline deviation (n = 31) were the most commonly reported complications. Maximum diameter of meningioma and its location significantly predicted the presence of perilesional oedema [χ2(2,47) = 6.03, P = .049, Nagelkerke R2 = 18.2%] and mass effect [χ2(2,71) = 16.73, P = .000, Nagelkerke R2 = 39.4%] in two logistic regression models. CONCLUSION: The probability of mass effect and perilesional oedema increased with the maximum diameter. Meningioma extending to both supratentorial and infratentorial compartments had the highest risk of having concomitant perilesional oedema and mass effect.

A rare case of Sweet syndrome secondary to melioidosis.

BACKGROUND: Melioidosis is an emerging infection in South Asia caused by Burkholderia pseudomallei with various clinical presentations that include pneumonia, bacteraemia, arthritis, and deep-seated abscesses. Various cutaneous manifestations have been described in association with melioidosis. However Sweet Syndrome secondary to melioidosis has not been reported in the literature. Herein we describe the first case of Sweet syndrome secondary to melioidosis. CASE PRESENTATION: A 53-year-old previously healthy Sri Lankan female presented with high-grade fever, painful oral ulcers, odynophagia and multiple bilateral cervical lymphadenopathies for 1 month. She also had a loss of appetite and weight. She had oral ulcers and bilateral blepharitis. Dermatological examination revealed multiple tender papules with a mamillated appearance and targetoid lesions with a yellowish centre over the face, upper trunk and upper limbs. She also had multiple tender subcutaneous nodules over the extensor aspect of upper limbs. Her inflammatory markers were significantly elevated. Aspirate from a submental lymph node abscess revealed the growth of Burkholderia pseudomallei. Melioidosis antibody titer was > 10,240. The histology of the skin lesions of the face and left forearm showed a prominent neutrophilic infiltrate in the dermis and the morphological features were in favour of Sweet syndrome with panniculitis. She was started on intravenous meropenem 2 g daily and showed rapid clinical improvement with the disappearance of skin lesions as well as a reduction in inflammatory markers. CONCLUSION: Sweet syndrome is an uncommon inflammatory disorder known to be associated with upper respiratory tract and gastrointestinal infections, malignancies and the use of certain drugs. Melioidosis is an emerging infection with various cutaneous manifestations. This is the first case of melioidosis causing the secondary sweet syndrome. It emphasizes the importance of considering melioidosis as a potential aetiology in patients with Sweet syndrome.

Kikuchi-Fujimoto disease associated with systemic lupus erythematosus complicated with hemophagocytic lymphohistiocytosis: a case report.

BACKGROUND: Kikuchi-Fujimoto disease, which was originally described in young women, is a benign condition characterized by necrotizing lymphadenitis and fever. Even though the clinical course is usually self-limiting, it can be associated with recurrences and rarely can be associated with systemic lupus erythematosus or can be complicated with hemophagocytic lymphohistiocytosis. We report the case of a 17-year-old Sri Lankan Sinhalese schoolboy who presented with fever and cervical lymphadenopathy diagnosed as Kikuchi-Fujimoto disease and was complicated with hemophagocytic lymphohistiocytosis subsequently. Later he fulfilled the criteria for systemic lupus erythematosus. CASE PRESENTATION: A 17-year-old previously healthy Sinhalese schoolboy presented with high-grade fever associated with chills and rigors associated with loss of appetite and loss of weight for more than 40 days. On examination, he had bilateral firm matted tender cervical lymphadenopathy and firm hepatomegaly. An excision biopsy of his right cervical lymph node revealed necrotizing lymphadenitis and immunohistochemistry of a lymph node biopsy favored Kikuchi disease. Initial antinuclear antibody and anti-double-stranded deoxyribonucleic acid tests were negative and his C3 and C4 levels were normal. An infections screening was negative. He was treated with steroids. While in hospital he developed hemophagocytic lymphohistiocytosis and renal impairment. Later his antinuclear antibody titer became positive in 1:160 and fulfilled the diagnostic criteria for systemic lupus erythematosus. He was managed with steroids and immune suppressive drugs and showed remarkable improvement. CONCLUSION: Although Kikuchi-Fujimoto disease is uncommon in male patients, it needs to be considered in patients with lymphadenopathy and fever. The disease can be complicated with hemophagocytic lymphohistiocytosis and the patients need continuous monitoring for the possible development of systemic lupus erythematosus later in the course.

A Rare Cause of Ascites-Disseminated TB with Peritonitis in a Middle-Aged Female.

BACKGROUND: Disseminated tuberculosis (TB) has been increasingly recognized in adults in the recent times due to increased prevalence of immune suppression. Here we describe a case of 47-year-old female who presented with disproportionate ascites where the diagnosis of disseminated TB was delayed. CASE REPORT: A 47-year-old previously healthy female presented with generalised body swelling with disproportionate ascites and loss of appetite and weight for four-month duration. She denied any contact or past history of TB and reported no respiratory symptoms. Physical examination revealed significant ascites. There was no lymphadenopathy or hepatosplenomegaly. Respiratory system examination was normal. Her Erythrocyte Sedimentation Rate (ESR) was above 100. Tuberculin skin test was positive with 17mm. Contrast Enhanced Computed Tomography (CECT) abdomen revealed chronic liver disease with ascites. Diagnostic laparoscopy was in favour of miliary TB and the peritoneal biopsy revealed granulomatous inflammation with caseous necrosis, suggestive of TB. The patient was started on antituberculosis treatment and subsequently improved. CONCLUSION: TB peritonitis due to disseminated TB should be considered in the differential diagnosis of disproportionate ascites. Even though the diagnosis is difficult, diagnostic laparoscopy and biopsy is very helpful. It is important to have an early diagnosis since delay in treatment can be detrimental in most cases.