Isoniazid-induced cerebellitis: a disguised presentation

Cerebellitis is a rarely encountered complication of isoniazid therapy. Its occurrence is usually associated with concomitant renal disease and haemodialysis. Herein, we report the case of a patient with this complication who presented with isolated bilateral symmetrical dentate nucleus T2 hyperintensities on magnetic resonance imaging. Isoniazid neurotoxicity has never been reported to cause bilateral dentate hyperintensities, for which the differentials are few and include metronidazole toxicity.     

Lymphocytic colitis complicated by a mass in the terminal ileum

Lymphocytic colitis is a chronic inflammatory disease affecting the bowel. The clinical course of lymphocytic colitis is believed to be benign with watery diarrhoea. We report herein what is, to the best of our knowledge, the first case of lymphocytic colitis complicated by a terminal ileal mass. A 23-year-old man presented with diarrhoea. Blind biopsies of samples taken from the terminal ileum, caecum and ascending colon showed features of lymphocytic colitis. He declined treatment with budesonide or 5-aminosalicylates. He presented 14 months later with pain over the right lumbar region and nausea. Computed tomographic enteroclysis showed a focal soft tissue enhancing mass at the terminal ileum. Excision of the soft tissue mass revealed that it was reactive nodular lymphoid hyperplasia with fibrous granulation tissue. In conclusion, an untreated lymphocytic colitis may result in the formation of an inflammatory mass lesion.     

Unusual presentation of adult xanthogranuloma

Xanthogranulomas are the most common form of non-Langerhans cell histiocytosis. Both adult and childhood forms have been described. Adult cutaneous forms can present as solitary or multiple yellowish, orange-red or tan-hued papules. Herein, we present the case of a 28-year-old Chinese man with a skin-coloured nodule on his left nasal ala that persisted for several months. While initial impression was that of a fibrous papule of the nose, the results of an excision biopsy showed histological features corresponding to xanthogranuloma. This case demonstrates the condition''s myriad of dermatological presentations, and adds to the differential diagnoses of a cutaneous lesion found in the head and neck region.     

Childhood autoimmune hepatitis in a paediatric unit of a tertiary care hospital

INTRODUCTION

Although childhood autoimmune hepatitis (AIH) has been extensively investigated in the West, data on AIH in the East is lacking. We aimed to investigate AIH’s clinical, biochemical and histological features, as well as its outcomes, in one of Singapore’s two major paediatric units.

METHODS

This was a retrospective study of children diagnosed with AIH in the paediatric unit of National University Hospital, Singapore, over the last 12 years. Children with de novo AIH after liver transplantation were excluded. The demographic and clinical features of the patients, and their laboratory, treatment and clinical outcomes were reviewed.

RESULTS

This study comprised ten patients (six females, four males), with a median age of 5.1 (range 2.1–13.8) years at diagnosis. Five patients had inflammatory bowel disease (IBD). Seven patients had type 1 AIH, and three had autoimmune sclerosing cholangitis (ASC) and IBD; none had type 2 AIH. The median level of aspartate aminotransferase at diagnosis was 183 (range 45–2,649) U/L. Prednisolone 1 mg/kg/day was prescribed at diagnosis for eight patients. Two patients were lost to follow-up and were treated symptomatically when they re-presented with end-stage liver disease. Azathioprine or mycophenolate mofetil was prescribed after 3–7 months of treatment. Normalisation of aminotransferase levels took an average of 5.3 (range 1–39) months.

CONCLUSION

AIH is a rare but important cause of liver pathology. Children in this region with elevated aminotransferases or unexplained hepatomegaly should be screened for AIH.     

Increased serum visfatin as a risk factor for atherosclerosis in patients with ischaemic cerebrovascular disease

INTRODUCTION

The present study aimed to investigate the possible associations between serum levels of visfatin, an adipokine, and atherosclerosis in patients with ischaemic cerebrovascular disease.

METHODS

A total of 95 participants were recruited for this study. Group A comprised 35 individuals with no history of cerebrovascular disease (control group) and Group B comprised 60 patients with ischaemic cerebrovascular disease. Group B was further categorised into two subgroups based on the ultrasonographic findings of the common carotid artery intima-media thickness (CCA-IMT) – Group B1 consisted of 21 patients with no atherosclerosis (i.e. CCA-IMT ≤ 0.9 mm) and Group B2 consisted of 39 patients with atherosclerosis (i.e. CCA-IMT > 0.9 mm). The body mass index, fasting blood total cholesterol, triglycerides, high-density lipoprotein cholesterol, low-density lipoprotein cholesterol and glucose levels of each patient were measured. Serum visfatin levels were determined using enzyme-linked immunosorbent assays. Visfatin levels were compared between groups, and stepwise logistic regression analysis was used to identify risk factors for atherosclerosis, including visfatin levels.

RESULTS

The mean serum visfatin level of the patients in Group B was higher than that in Group A (75.5 ± 77.80 ng/mL vs. 8.6 ± 4.69 ng/mL; p < 0.05) and the level was higher in patients from Group B2 than those from Group B1 (89.0 ± 80.68 ng/mL vs. 50.4 ± 72.44 ng/mL; p < 0.05). Multivariate regression analysis showed that CCA-IMT values were not significantly associated with visfatin levels. However, logistic regression analysis showed that serum visfatin was an independent risk factor for atherosclerosis (odds ratio 37.80; p = 0.004).

CONCLUSION

Serum visfatin may be an independent risk factor for cerebral infarction, as high serum visfatin levels are positively associated with the underlying pathogenic mechanisms of ischaemic cerebrovascular disease.     

Distribution of branchial anomalies in a paediatric Asian population

INTRODUCTION

The objective of the present study was to review the distribution and incidence of branchial anomalies in an Asian paediatric population and highlight the challenges involved in the diagnosis of branchial anomalies.

METHODS

This was a retrospective chart review of all paediatric patients who underwent surgery for branchial anomalies in a tertiary paediatric hospital from August 2007 to November 2012. The clinical notes were correlated with preoperative radiological investigations, intraoperative findings and histology results. Branchial anomalies were classified based on the results of the review.

RESULTS

A total of 28 children underwent surgery for 30 branchial anomalies during the review period. Two children had bilateral branchial anomalies requiring excision. Of the 30 branchial anomalies, 7 (23.3%) were first branchial anomalies, 5 (16.7%) were second branchial anomalies, 3 (10.0%) were third branchial anomalies, and 4 (13.3%) were fourth branchial anomalies (one of the four patients with fourth branchial anomalies had bilateral branchial anomalies). In addition, seven children had 8 (26.7%) branchial anomalies that were thought to originate from the pyriform sinus; however, we were unable to determine if these anomalies were from the third or fourth branchial arches. There was inadequate information on the remaining 3 (10.0%) branchial anomalies for classification.

CONCLUSION

The incidence of second branchial anomalies appears to be lower in our Asian paediatric population, while that of third and fourth branchial anomalies was higher. Knowledge of embryology and the related anatomy of the branchial apparatus is crucial in the identification of the type of branchial anomaly.     

The clinical presentation and diagnosis of ketamine-associated urinary tract dysfunction in Singapore

Ketamine is a short-acting anaesthetic agent that has gained popularity as a ‘club drug’ due to its hallucinogenic effects. Substance abuse should be considered in young adult patients who present with severe debilitating symptoms such as lower urinary tract symptoms, even though the use of controlled substances is rare in Singapore. Although the natural history of disease varies from person to person, a relationship between symptom severity and frequency/dosage of abuse has been established. It is important to be aware of this condition and have a high degree of clinical suspicion to enable early diagnosis and immediate initiation of multidisciplinary and holistic treatment. A delayed diagnosis can lead to irreversible pathological changes and increased morbidity among ketamine abusers.     

Prolonged hepatitis and jaundice: a rare complication of paediatric Epstein-Barr virus infection

We herein report the case of a 14-year-old girl with Epstein-Barr virus (EBV) infectious mononucleosis who developed prolonged hepatitis and jaundice. At presentation, she had tender hepatomegaly with a markedly deranged liver function test. Abdominal ultrasonography showed hepatomegaly and a thickened gallbladder wall. During the subsequent 11 weeks, her transaminases showed two further peaks, which corresponded with clinical deterioration. Her highest alanine transaminase level was 1,795 µ/L and total bilirubin level was 154 µmol/L. She recovered fully with conservative management. EBV-related liver involvement is typically mild and self-limiting. We believe that tender hepatomegaly and gallbladder thickening may be important predictors of significant liver involvement. Although multiple transaminase peaks may occur, we do not consider this an indication for antiviral or immunosuppressive therapy. In the absence of strong evidence supporting the use of any specific therapy, we recommend a conservative approach for an immunocompetent patient.     

阿格列汀治疗三硝基苯磺酸诱导的溃疡性结肠炎小鼠的实验研究

目的观察DPP-4抑制剂Alogliptin对三硝基苯磺酸诱导的溃疡性结肠炎模型小鼠结肠的MPO值,血清IL-8、TNF-α和GLP-2水平的影响,并探讨Alogliptin对炎症性肠病(IBD)的治疗机制。方法雄性BALB/c小鼠48只,分为6组,每组8只,分别为正常对照组、模型组、柳氮磺吡啶治疗组(520 mg/kg),Alogliptin高、中、低剂量组(1、0.3、0.1mg/kg)。模型组采用三硝基苯磺酸(TNBS)对BALB/c小鼠进行灌肠,制作溃疡性结肠炎小鼠模型,经灌胃给予药物治疗后,观察DPP-4酶抑制剂Alogliptin对肠炎小鼠腹泻,组织形态损伤,结肠组织MPO酶活力,血清IL-8、TNF-α和GLP-2含量的影响。结果与模型组相比,Alogliptin高、中剂量组能够改善肠炎小鼠临床症状,减轻结肠黏膜损伤,显著降低TNBS诱导的溃疡性结肠炎小鼠的DAI评分(P<0.01),并且治疗组小鼠结肠组织MPO酶活性、血清IL-8和TNF-α的浓度较模型组显著降低(P<0.05);而Alogliptin治疗组小鼠血清GLP-2的含量较模型组和正常组都有显著升高(P<0.05)。结论 DPP-4酶抑制剂Alogliptin能够有效抑制小鼠肠道炎性细胞的浸润,减轻肠黏膜的病理性损伤,对TNBS诱导的肠炎小鼠具有显著的治疗作用。     

Extracorporeal life support for cardiac arrest in a paediatric emergency department

The initiation of extracorporeal membrane oxygenation (ECMO) in the emergency department (ED) is a rare event. Herein, we report a case of acute fulminant myocarditis in a nine-year-old girl who was successfully resuscitated by early initiation of ECMO support in the paediatric ED of KK Women’s and Children’s Hospital, Singapore. The patient had rapidly progressed into a witnessed pulseless ventricular tachycardia on presentation, and ECMO was started in the ED following the failure of standard resuscitation measures to establish spontaneous circulation. ECMO was continued for nine days. The patient recovered well with normal neurocognitive function. The initiation of ECMO in the ED is potentially life-saving in the resuscitation of children with witnessed in-hospital cardiac arrest due to a reversible cause.     

An unusual presentation of recurrent uterine rupture during pregnancy

We describe a case of recurrent uterine rupture at the site of a previous rupture. Our patient had a history of right interstitial pregnancy with spontaneous uterine fundal rupture at 18 weeks of pregnancy. During her subsequent pregnancy, she was monitored closely by a senior consultant obstetrician. The patient presented at 34 weeks with right hypochondriac pain. She was clinically stable and fetal monitoring showed no signs of fetal distress. Ultrasonography revealed protrusion of the intact amniotic membranes in the abdominal cavity at the uterine fundus. Uterine rupture is a rare but hazardous obstetric complication. High levels of caution should be exercised in patients with a history of prior uterine rupture, as they may present with atypical symptoms. Ultrasonography could provide valuable information in such cases where there is an elevated risk of uterine rupture at the previous rupture site.     

An unusual presentation of atrial myxoma

Myxomas are uncommon primary cardiac tumours that usually affect the left atrium. We herein report the case of a patient who presented with right heart failure and proteinuria, leading to the diagnosis of atrial myxoma. Surgical resection resulted in resolution of the patient’s symptoms.     

Clinical presentation of inflammatory bowel disease: a hospital based retrospective study of 379 patients in eastern China

Background Numerous studies from Europe and North America have provided a weahh of information regarding the epidemiological and clinical characteristics of inflammatory bowel disease (IBD) in Caucasians. Previous studies in mainland China have been limited by small patient numbers or by lack of detailed information about clinical subgroups of the disease. This study was carried out to assess the demographic and clinical characteristics of IBD in Chinese patients. Methods In the Sir Run Run Shaw Hospital between 1994 and 2003, 379 patients were diagnosed as IBD. Demographic and clinical data were collected and analysed. Results Of 379 patients, 317 had ulcerative colitis (UC) (83.6% , 168 male, 149 female, male-female ratio 1.13: 1, age range at diagnosis 14-79 years, mean age 44 years) and 62 had Crohn‘s disease (CD) (16.4% , 39 male and 23 female, male-female ratio 1.70:1 , age range at diagnosis 13 -70 years, mean age 33 years). In UC, 11.4% of patients had proctitis, 25.2% had proctosigmoiditis, 18.6% were diseased to the splenic flexure and 44. 8% had extensive colitis. Nine patients with UC (2.8%) had arthritis, three patients (0.9%) had iritis or conjunctivitis. Of the 62 CD patients, 16 (25.8%) had diseases restricted to the terminal ileum; 15 (24.2%) had colonic diseases; 20 (32.3%) had ileocolonic disease and 11 (17.7%) had disease involving the upper gastrointestinal tract. Conclusions This study shows similar caracteristics of IBD to that in the West but there are some differences with respect to severity and extraintestinal manifestations. The ethnic and geographic differences may give important clues to the aetiology of IBD.     

Achalasia and Down syndrome: a unique association not to be missed

Achalasia is a rare primary oesophageal motility disorder that presents as a functional obstruction at the oesophago-gastric junction. The prevalence of achalasia in Down syndrome is much higher, which implies a unique association between these two uncommon conditions. Although the exact aetiology of achalasia is unknown, studies have proposed that its pathogenesis is related to autoimmune, infectious or genetic factors, leading to the intrinsic loss of inhibitory myenteric neurons in both the oesophagus and lower oesophageal sphincter. We herein report the case of a 16-month-old girl with Down syndrome and achalasia who was initially treated for gastro-oesophageal reflux disease. The diagnosis of achalasia was made only when her condition deteriorated, with subsequent failure to thrive, and upon further investigations, including barium swallow study and upper endoscopy. We also review the various mechanisms postulated in the development of achalasia in Down syndrome, as well as the various treatment modalities available for this rare disorder.     

Recurrent and persistent pityriasis rosea: an atypical case presentation

We report a case of atypical pityriasis rosea in a 24-year-old Malay man. He presented with an 11-month history of three recurrent and persistent episodes of pityriasis rosea associated with oral ulcers. The first episode lasted for one month and recurred within 14 days. The second episode lasted for three months and recurred within nine days. The third episode lasted for seven months. Although all three episodes were not preceded by any prodromal symptoms, a herald patch was noted on three different sites (the left iliac fossa, abdomen and chest) on each successive episode. Recurrent pityriasis rosea and its association with oral ulcers, although quite uncommon, have been reported in the literature. However, reports of multiple recurrences, with prolonged duration of each episode and very short remissions in between, have not been made. To the best of our knowledge, this is the first report of such unique presentation.     

Infant lumbar and thoracic epidurals for abdominal surgeries: cases in a paediatric tertiary institution

INTRODUCTION

There is strong evidence that epidural analgesia provides good postoperative pain relief in adults, but its use in infants is less established. In this retrospective study, we present our experience with managing infant epidural analgesia for abdominal surgeries in a tertiary paediatric institution.

METHODS

The records of 54 infants who had received a thoracic or lumbar epidural as perioperative analgesia for abdominal surgeries were included. The mean age of the infants was 6.1 (standard deviation [SD] 3.8) months and their mean weight was 6.8 kg (SD 1.8). Most (63%) had an ASA (American Society of Anesthesiologists) status of 2 and all underwent elective gastrointestinal, urogenital, hepatobiliary or retroperitoneal surgeries. 20 catheters (37.0%) were inserted in the thoracic region and 33 (61.1%) in the lumbar region.

RESULTS

A total of 52 (96.3%) catheters provided adequate intraoperative analgesia and 36 (66.7%) provided effective analgesia for the postoperative period. Active management of epidural analgesia, such as through epidural top-ups and infusion rate adjustment, was necessary to optimise analgesia in 22 (44%) of the 50 patients postoperatively. Reasons for premature catheter removal were mainly technical issues such as catheter disconnection, leakage and blockage.

CONCLUSION

Our data suggests that in experienced hands, specialised settings and active management, the success rate of epidural analgesia in infants undergoing major abdominal surgeries is high and without major incident.     

Clinical profile and visual outcome of traumatic paediatric cataract in suburban Malaysia: a ten-year experience

INTRODUCTION

Available data on traumatic cataract in Asian children is primarily confined to South Asian countries. We aimed to describe the demographics, nature of injury and visual outcomes of Malaysian children with traumatic cataract from a suburban area, and discuss the literature on Asian children with this condition.

METHODS

We conducted a retrospective study of 29 children below 17 years of age who were diagnosed with traumatic paediatric cataract and who attended Hospital Universiti Sains Malaysia, Kelantan, Malaysia, between January 2000 and December 2010. Follow-up periods ranged from 12 to 120 months. Demographic data, clinical features, mechanism and extent of injury, and final visual outcome were recorded.

RESULTS

The study population was predominantly male. The right eye was injured in 62.07% of patients. A majority of patients had penetrating injuries, with the most common cause being injury by an organic foreign body (24.14%). Presenting visual acuity worse than 6/60 was observed in 68.97% of patients. Only 34.48% of patients had a final corrected visual acuity of 6/12 and better. 55.18% of patients were operated on within less than one month of their injuries. A majority of children sustained concurrent injuries to the anterior segment structures. Corneal opacity and amblyopia were the most common causes of poor final visual acuity.

CONCLUSION

Health education and awareness are essential tools that can prevent avoidable blindness due to traumatic cataract in the paediatric population. The importance of rehabilitation programmes for these patients should be emphasised.     

Hirayama disease in a 17-year-old Chinese man

Hirayama disease is an uncommon cervical myelopathy associated with neck flexion. It has been postulated to be related to the anterior shifting of the posterior dura of the lower cervical dural canal during neck flexion, resulting in lower cervical cord atrophy with asymmetric flattening. We report a case of Hirayama disease in a 17-year-old Chinese man and demonstrate the use of dynamic flexion magnetic resonance imaging of the cervical spine in the diagnosis of the disease.     

Performance of the CKD-EPI creatinine-cystatin C glomerular filtration rate estimation equations in a multiethnic Asian population

INTRODUCTION

Clinical practice guidelines recommend using creatinine-based equations to estimate glomerular filtration rates (GFRs). While these equations were formulated for Caucasian-American populations and have adjustment coefficients for African-American populations, they are not validated for other ethnicities. The Chronic Kidney Disease-Epidemiology Collaborative Group (CKD-EPI) recently developed a new equation that uses both creatinine and cystatin C. We aimed to assess the accuracy of this equation in estimating the GFRs of participants (healthy and with chronic kidney disease [CKD]) from a multiethnic Asian population.

METHODS

Serum samples from the Asian Kidney Disease Study and the Singapore Kidney Function Study were used. GFR was measured using plasma clearance of ^99mTc-DTPA. GFR was estimated using the CKD-EPI equations. The performance of GFR estimation equations were examined using median and interquartile range values, and the percentage difference from the measured GFR.

RESULTS

The study comprised 335 participants (69.3% with CKD; 38.5% Chinese, 29.6% Malays, 23.6% Indians, 8.3% others), with a mean age of 53.5 ± 15.1 years. Mean standardised serum creatinine was 127 ± 86 µmol/L, while mean standardised serum cystatin C and mean measured GFR were 1.43 ± 0.74 mg/L and 67 ± 33 mL/min/1.73 m², respectively. The creatinine-cystatin C CKD-EPI equation performed the best, with an estimated GFR of 67 ± 35 mL/min/1.73 m².

CONCLUSION

The new creatinine-cystatin C equation estimated GFR with little bias, and had increased precision and accuracy in our multiethnic Asian population. This two-biomarker equation may increase the accuracy of population studies on CKD, without the need to consider ethnicity.     

High-grade culprit lesions are a common cause of ST-segment elevation myocardial infarction

INTRODUCTION

Conventional knowledge holds that the majority of ruptured atherosclerotic plaques causing ST-segment elevation myocardial infarction (STEMI) are found in moderate stenoses that produce < 50% loss of arterial diameter. This study aimed to analyse the culprit lesions in patients who presented with STEMI and underwent primary percutaneous coronary intervention (PPCI) at our institution.

METHODS

Patients who underwent PPCI between June 2008 and August 2010 at our institution were included in the analysis. Quantitative coronary angiography was performed for the culprit lesions immediately after antegrade flow was restored by thrombectomy, low-profile balloon predilatation or guidewire crossing.

RESULTS

A total of 1,021 patients were included in the study. The mean age was 57 ± 12 years and 85.2% were male. Lesion measurement was done after coronary flow was restored by thrombectomy (73.1%), balloon dilatation (24.1%) and following guidewire passage across the lesion (2.8%). Mean minimal luminal diameter was 1.1 ± 0.5 mm, mean reference vessel diameter was 2.8 ± 0.6 mm, mean diameter stenosis was 61 ± 16% and mean lesion length was 16 ± 6 mm. Most (80.2%) of the culprit lesions had diameter stenoses > 50% (p < 0.01). Although balloon angioplasty was performed in 24.1% of the patients, the majority (64.2%) still had diameter stenoses > 50%. High-grade stenoses (> 50%) were more frequently observed in male patients (p = 0.04).

CONCLUSION

Contrary to the existing paradigm, we found that most of the patients with STEMI in our institution had culprit lesions with diameter stenosis > 50%.