【摘要】 目的：总结Alport综合征的临床表现，尤其是眼部特征。方法：回顾性分析32例被确诊为Alport综合征患者的内科、耳鼻喉科和眼科检查结果。结果：患者30例(93.7%)有疾病家族史。所有患者均有不同程度的肾脏病变：18例(56.3%)有肾功能衰竭，4例(12.5%)肾功能不全，10例(31.3%)血尿。患者20例(62.5%)有感音神经性耳聋。患者13例(40.6%)有眼部异常表现，其中5例(15.6%)为典型性改变：前圆锥晶体3例，黄斑周围斑点2例。结论：眼部异常不是Alport综合征诊断的必需条件，但因其典型的眼科表现应当引起眼科医师的注意，以便早期诊断治疗。
【关键词】 Alport综合征;前圆锥晶状体;黄斑斑点
Bilateral endogenous endophthalmitis in a young immunocompetent lady with septic arthritis
Nurhamiza Buang, Shawarinin Jusuh, Bakiah Shaharuddin, Shatriah Ismail
Department of Ophthalmology, School of Medical Sciences, Universiti Sains Malaysia, 16150 Kubang Kerian, Kelantan, Malaysia.
Abstract A 20yearold healthy lady was admitted for septic arthritis in the right knee. Two weeks after the onset of joint pain, she complained progressive blurring of vision in both eyes. Slitlamp examination revealed evidence of inflammation in both anterior and posterior segments. There were multiple chorioretinal lesions on both fundi. Synovial fluid aspiration of the right knee and blood culture grew Staphylococcus aureus. She responded well to topical and systemic antibiotics. Her final visual acuity was 6/6 in both eyes. This case illustrates a rare presentation of bilateral endogenous endophthalmitis in an immunocompetent young patient presenting with a concurrent septic arthritis. Early diagnosis and a prompt management carry a favorable visual prognosis.
KEYWORDS: septic arthritis; endogenous endophthalmitis; immunocompetent
INTRODUCTION
Endogenous endophthalmitis is relatively a rare condition but carries severe and potentially blinding ocular infection. It is caused by a variety of infectious agents, namely bacteria or fungal that reaches the eye from the primary site via a haematogenous spread. Meningitis, endocarditis, infection of urinary or gastrointestinal tract and wound are the most common causes of endogenous endophthalmitis, while other less common source includes pharyngitis, pneumonia, pyelonephritis and intraabdominal abscess [1]. Septic arthritis is an uncommon primary source of infection [2]. We report a case of bilateral endogenous endophthalmitis with septic arthritis of right knee as the primary source of infection in a young immunocompetent lady. Early diagnosis and prompt management is essential to preserve a favorable visual outcome in this uncommon condition.
CASE REPORT
A 20yearold immunocompetent lady was admitted for swelling in the right knee and fever for two weeks duration. She was diagnosed as septic arthritis in the right knee and started on intravenous Cloxacillin 500mg 6 hourly and intravenous crystalline penicillin 1.2 MU 6 hourly. She underwent synovial fluid aspiration of the right knee on the third day of admission.
One day later, she complained blurred vision in both eyes. It was associated with mild discomfort and redness. However, there was no history of trauma, skin disease, underlying medical illness or consumption of immunosuppressive medications. Visual acuity was 6/24 in both eyes. Both conjunctivas were injected. Slitlamp examination of both anterior and posterior segments revealed moderate inflammation. There were multiple small whitish chorioretinal lesions on both fundi. Systemic examination revealed a lethargic and febrile patient. Her right knee was swollen, warm and tender in flexion position. Other joints and systemic examinations were unremarkable. There was no evidence of osteomyelitic changes or gas shadow on the Xray of right knee. Synovial fluid aspiration of the right knee revealed numerous pus cells. Both synovial fluid and blood cultures grew Staphylococcus aureus. Vitreous tap and injection of intravitreal antibiotic were refused by the patient. Full blood count revealed leukocytosis with raised ESR at 91mm/first hour. Collagen screenings and other infective workups were negative.
She was prescribed gutt ciprofloxacin 2 hourly, gutt Pred Forte 1.5% 6 hourly and gutt tropicamide 8 hourly in both eyes, and added on to intravenous ciprofloxacin 200mg 12 hourly. She showed a dramatic clinical improvement a week later (Figure 1). The intravenous antibiotics were completed for two weeks, while oral Cloxacillin was maintained for another one month. Her final visual acuity was 6/6 in both eyes.Table 2Summary of visual outcome in endogenous endophthalmitis related to septic arthritis reported in the literature 19962009(略)
DISCUSSION
Septic arthritis denotes as a primary source or concomitant site of infection in endogenous endophthalmitis. It accounts 8% of endogenous endophthalmitis in 2 large case series [1,2]. Table 1 illustrates the patients risk factors and potential microorganism causing endogenous endophthalmitis. Table 2 summarizes our MEDLINE literature search on endogenous endophthalmitis and septic arthritis from 1996 to 2009. A total of 8 patients were reported in MEDLINE literature on endogenous endophthalmitis and septic arthritis from 1996 to 2009, who fulfilled our inclusion criteria, in the past 13 years [25]. Of these 10 eyes, Group B Streptococcus was the main causative microorganism in 6 eyes(60.0%). The remaining were Streptococcus pyogenes (one eye), Streptococcus
pneumonia (one eye) and Staphylococcus aureus (two eyes). Our patient is the only patient who developed septic arthritis due to a Staphylococcus sp. microorganism. There was no fungal etiology observed causing septic arthritis in our analysis. These patients age ranged from 53 to 82 years old. 2 patients had diabetes mellitus and 1 patient had history of splenectomy for more than 50 years [2,4]. 7 of these patients (70.0%) presented with profound visual impairment in the affected eyes. There was a trend of poor final visual acuity in all patients infected with Streptococcus sp, worst in Group B
Streptococcus. However due to small sample size, we are unable to further analyze the association between type of microorganism and final visual outcome. Our patient was the youngest patient in this analysis. She presented with moderate visual impairment but ultimately, her final visual outcome in both eyes was satisfactory. Despite early diagnosis and prompt treatment, we postulate that immunocompetent status and low virulent microorganism explain her good recovery.
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