A rare asymptomatic metallic intraocular foreign body retained in the anterior chamber for 15 years: A case report

Rationale:Intraocular foreign bodies (IOFBs) are common in ocular injuries, but asymptomatic metallic IOFBs retained in the anterior chamber for years are rare.Patient concerns:A 31-year-old female presented with blurred vision in her right eye after lumbar magnetic resonance imaging. Her best-corrected vision acuity was 0.6 in the right eye and 1.0 in the left eye. Slit-lamp examination revealed a brown granular foreign body in the anterior chamber and pigmentation of the limbus. Lens and retina examination indicated ocular siderosis. Corneal endothelioscopy revealed decreased endothelial cell density. A detailed history showed ocular globe injury 15 years earlier.Diagnoses:Anterior chamber IOFB with ocular siderosis.Interventions:Anterior chamber foreign body removal was performed with appropriate incision and forceps.Outcomes:The anterior chamber IOFB was successfully removed and examined as a magnetic metal foreign body. The best-corrected vision acuity was 1.0 at 1 day postoperatively. An abnormal electroretinogram with a 12% decrease in the “b” wave and a 91% decrease in the “a” wave was observed 3 months postoperatively. There were no intraoperative or postoperative complications during a 3-month follow-up.Lessons:Eye trauma should be examined carefully to exclude IOFBs. Asymptomatic anterior chamber foreign bodies can also cause corneal endothelial injury and ocular siderosis. Careful examination and timely management are needed in such cases.     

Rapid progression of high-risk proliferative diabetic retinopathy induced by insulin intensive therapy: A case report

Rationale:To summarize and analyze a case of rapid progression of high-risk proliferative diabetic retinopathy after the insulin intensive therapy (IT).Patient concerns:A 58-year-old type 2 diabetes female patient suffered a rapid and dramatic decline of vision acuity in the left eye in 2 months after the insulin IT. However, the best corrected visual acuity (BCVA) of her right eye, which was in much severer condition and received panretinal photocoagulation (PRP) before, improved after the IT.Interventions:The patient received intravitreal injection of conbercept (IVC) to her left eye, and 5 days later, underwent pars plana vitrectomy (PPV), combined with PRP and silicon oil injection.Outcomes:The postoperative BCVA of the left eye was 20/200 and improved to 20/160 one month later. During the subsequent 2 months of follow-up, her BCVA remained 20/160 in both eyes. Her blood glucose level also remained stable.Lessons:Insulin IT for untreated proliferative diabetic retinopathy (PDR) patients can cause severe and irreversible consequences, so for such patients, the conservative treatment for glycemic control may be much safer. But if insulin IT is inevitable, the patient should undergo PRP promptly before the IT, and close eye monitoring during the IT is also essential.     

Choroidal neovascularization secondary to half-dose photodynamic therapy for chronic central serous chorioretinopathy: A case report

Rationale:Half-dose or reduced-fluence photodynamic therapy (PDT) with verteporfin has been well acknowledged to be the most effective and permanent treatment with very low rates of complications. However, we report a case of chronic central serous chorioretinopathy (CSC) who developed choroidal neovascularization (CNV) secondary to half-dose PDT within only 3 weeks. Such an occurrence following this short a course of treatment has not been reported previously.Patient concerns:A 46-year-old Chinese man who had been diagnosed as acute more than 1 year ago revisited our department recently and complained of blurred vision again in his left eye.Diagnoses:Fluorescein fundus angiography (FFA) and indocyanine green angiography (ICGA) revealed patchy hyperfluorescent dots and optical coherence tomography (OCT) indicated irregular flat pigment epithelium detachment (PED) in the central macula. The patient was diagnosed with chronic CSC.Interventions:The patient was treated by half-dose PDT with verteporfin. Three weeks later, the patient complained of sudden blurred vision and fundus examination showed macular hemorrhages with a best-corrected visual acuity (BCVA) of 20/250. OCT angiography (OCTA) showed a distinct area of flower-like CNV located within the deep retinal slab. Secondary CNV had developed after a quite short course of half-dose PDT treatment. Subsequently, the patient was administered by 2 intravitreal injections of aflibercept (2 mg).Outcomes:Two months after the second intravitreal injection, macular hemorrhages and secondary CNV were completely resolved, and the BCVA improved to 20/25.Lessons:Patients of chronic CSC with irregular PED who undergo PDT should be warned of secondary CNV within a short course after treatment. If happened, it should be treated by intravitreal injections of anti-vascular endothelial growth factor agents as soon as possible.     

Chronic endogenous fungal endophthalmitis: diagnostic and treatment challenges: A case report

Rationale:Endogenous fungal endophthalmitis (EFE) is a sight-threatening complication of systemic fungemia. As the prevalence rises, treatment remains a challenge especially when there is a failure in first-line treatment or drug-resistant fungus. This case report studies a case of chronic EFE, focusing on the diagnostic procedures, treatment options, monitoring parameters and the treatment outcomePatient concerns:A 64-year-old man with underlying well controlled diabetes mellitus was treated with 2 weeks’ course of intravenous antifungal fluconazole for pyelonephritis as his blood culture grew Candida albicans. Concurrently, he complained of 3 months of bilateral painless progressive blurring of vision. At presentation, his visual acuity (VA) was light perception both eyes. Ocular examination revealed non granulomatous inflammation with dense vitritis of both eyes.Diagnosis:He was diagnosed with EFE but the condition responded poorly with the medications.Interventions:He was treated with intravitreal (IVT) amphotericin B and fluconazole was continued. Vitrectomy was performed and intraoperative findings included bilateral fungal balls in the vitreous and retina with foveal traction in the left eye. Postoperatively, vision acuity was 6/24, N8 right eye and 2/60, N unable for left eye with extensive left macular scar and hole. Vitreous cultures were negative. He received multiple IVT amphotericin B and was started on topical steroid eye drops for persistent panuveitis with systemic fluconazole. Ocular improvement was seen after switching to IVT and topical voriconazole. Despite this, his ocular condition deteriorated and he developed neovascular glaucoma requiring 3 topical antiglaucoma agents. Panretinal photocoagulation was subsequently performed.Outcomes:At 3 months’ follow-up, his vision acuity remained at 6/24 for right eye and 2/60 for the left eye. There was no recurrence of inflammation or infection in both eyes.Lessons:Voriconazole could serve as a promising broad spectrum tri-azole agent in cases of failure in first-line treatment or drug-resistant fungus.     

Repeated rotation of a toric implantable collamer lens: A case report

Introduction:Implantable collamer lens have been used widely worldwide, and have been accepted by more and more doctors and patients due to good safety, stability, and effectiveness. However, there is still a problem of crystal rotation. The large angle rotation (over 10°) would weaken the original astigmatism correction effect and even induce irregular astigmatism, seriously affecting the visual quality of patients. Herein, we reported a case who had 2 times of crystal rotations after toric implantable collamer lens (TICL) implantation.Patient concerns:The patient was a 38-year-old man who underwent TICL implantation for the correction of high myopic astigmatism in eyes. He presented a sudden decrease in the visual acuity (VA) of the left eye 4 months after the TICL implantation. The uncorrected visual acuity (UCVA) was 8/20 (refraction, +2.25 −5.25 × 68).Diagnosis:Rotation of TICL was diagnosed. The toric marks with a rotation of 75° counter-clockwise from the original position were observed.Interventions:The TICL was re-set to the original position, leading to the UCVA of 12/20 in the left eye (refraction, −0.00 −0.75 × 131), with the vaulting of 589 μm. Ten months after the TICL relocation, the patient again presented a sudden decrease in the VA of the left eye, with the UCVA of 2/20 (refraction, +2.25 −5.00 × 66). Again, the toric marks with a rotation of 75° counter-clockwise from the original position was observed, just at the same position as the last rotation. This time, the TICL was removed. The axis and power were recalculated, and a new TICL was implanted, with the rotation of 73° counter-clockwise from the horizontal line of the temporal side.Outcomes:The patient obtained a final UCVA of 12/20 in the left eye (refraction, +0.50 −0.50 × 26), which remained stable in the 6-month follow-up period, with the vaulting of 602 μm.Lessons:Rotation is a common complication after TICL surgery. Relocation or replacement of TICL are safe and efficient ways to recover VA due to TICL rotation.     

Drug induced oral erythema multiforme: Case report

Introduction:Drug induced oral erythema multiforme a rare clinical entity which involves only the lips and oral mucosa without skin involvement. These lesions are difficult in diagnosing with other oral ulcerative lesions with similar clinical manifestations.Patient concerns:This article presents 2 case reports of Oral erythema multiforme in which drugs were the precipitating factor. Its etiopathogenesis, differential diagnosis and treatment modalities of the disease is discussed.Diagnosis:Based on patient''s complaints, drug history and clinical appearance, provisional diagnosis of drug induced erythema multiforme was considered.Intervention:For case 1, patient was instructed to discontinue usage of drug and prescribed systemic steroid (Prednisolone 10 mg/d) for a week along with germicidal drugs to prevent secondary infection. Medication was tapered to 5 mg/d after first week.For case 2, patient was instructed to discontinue the drug and systemic steroid prednisolone 20 mg /d for 1 week with tapering dose of 10 mg/d for the second week was administered.Outcome:For case 1 and case 2 healing of the lesions were evident on third week of follow up.Conclusion:Medications should be taken under medical supervision. Over the counter drugs might lead to allergic reactions like drug induced oral erythema multiforme, which is a rare variant and needs to be differentiate from other oral ulcerative lesion for prompt management and follow-up.     

Myelin oligodendrocyte glycoprotein antibody-associated optic neuritis in a COVID-19 patient: A case report

Rationale:Coronavirus disease 2019 (COVID-19) has spread worldwide. It involves multiple organs of infected individuals and encompasses diverse clinical manifestations. We report a case of acute optic neuritis (ON) associated with myelin oligodendrocyte glycoprotein (MOG) antibody possibly induced by COVID-19.Patient concerns:A 47-year-old man presented to our clinic with left eye pain and vision loss. Magnetic resonance imaging of the orbit revealed the bilateral high intensity of the optic nerve sheaths. He tested positive for COVID-19 by polymerase chain reaction (PCR) testing on the day of admission but he had no signs of respiratory illness. Laboratory testing revealed that MOG immunoglobulin G (MOG IgG) was positive, but other antibodies including aquaporin-4 were negative.Diagnosis:The patient was diagnosed with MOG antibody-positive acute ON possibly induced by COVID-19.Interventions:Steroid pulse therapy consisting of methylprednisolone 1 g/day for a total of 3 days, followed by an oral prednisolone taper was performed.Outcomes:His left eye pain was immediately relieved, and his decimal vision improved from 0.03 to 0.1 on the day of discharge. Outpatient follow-up 2 weeks later revealed left a decimal vision of 1.2, and a complete resolution of the left eye pain.Lessons:Our case indicated that COVID-19 might trigger an autoimmune response that leads to MOG antibody-associated ON, similar to other pathogens that were reported in the past. The treatment response to steroid pulse therapy was preferable following a typical course of MOG antibody-positive ON.     

Neuromyelitis optica spectrum disorder with active replication of hepatitis B virus and seropositive anti-aquaporin-4 antibody: A case report

Rationale:Neuromyelitis optica spectrum disorder (NMOSD) associated with active replication of hepatitis B virus (HBV) is rare. High-dose corticosteroids are the mainstay treatment of NMOSD; however, these may cause reactivation of viral replication in patients with stable HBV which may lead to liver damage. Therefore, care should be placed in corticosteroid use in patients with NMOSD and HBV infection.Patient concerns:Herein, we report the case of a 31-year-old woman with NMOSD and HBV infection who was seropositive for anti-aquaporin-4 antibody. The stable and HBV carrier status of the patient led to the deferment of antiviral and hepatoprotective agents in early treatment. However, liver function impairment was detected during follow-up, with an improvement in the best-corrected visual acuity.Diagnoses:The patient was diagnosed with NMOSD with active replication of HBV and seropositive anti-aquaporin-4 antibody considering the medical history and ancillary examinations.Interventions:To manage NMOSD, intravenous high-dose methylprednisolone (20 mg/kg d) was administered for 5 days which was gradually tapered to oral steroids. However, liver function impairment was observed during follow-up; therefore, anti-HBV drugs (entecavir) and hepatoprotective drugs (bicyclol or polyunsaturated phosphatidylcholine) were administered.Outcomes:A marked improvement was observed in the patient''s best-corrected visual acuity after 4 weeks of treatment. However, follow-up examinations revealed liver function damage which necessitated administration of antiviral and hepatoprotective drugs. Liver function normalized after 1 month.Lesson:This case underscores the importance of preventive treatment of liver protection in patients with HBV infection prior to or simultaneous with glucocorticoid therapy and furthermore, there is an urgent need to develop authoritative guidelines regulating corticosteroid use in the treatment of patients with HBV infection.     

Low vision rehabilitation in improving the quality of life for patients with impaired vision: A systematic review and meta-analysis of 52 randomized clinical trials

Background & aim:Low vision rehabilitation optimizes the use of residual vision after severe vision loss, but also teaches skills to improve visual functioning in daily life. These skills promote independence and active participation in society. This meta-analysis was designed to evaluate the efficacy of low vision rehabilitation in improving the quality of life (QoL) in visually impaired adults.Methods:We searched the Cochrane Library, PubMed, EMBASE, and Web of Science up to January 1, 2020. Randomized controlled trials (RCTs) that compared rehabilitation interventions with active or inactive controls were included. The standardized mean difference (SMD) with a 95% confidence interval (CI) was estimated to compare outcomes. Two reviewers extracted data and assessed trial quality independently. All statistical analyses were performed using the standard statistical procedures of RevMan 5.2.Results:A total of 52 RCTs with 6,239 participants were included in this meta-analysis. Compared to inactive comparators including waiting list or no care, low vision rehabilitation improved vision-related QoL, visual functioning (QoL: psychological aspect), and self-efficacy or self-esteem (QoL: psychological aspect), with pooled SMDs of −0.61 (95% CI −0.95 to −0.26; P = .0006), -1.14 (95% CI −1.69 to −0.59; P < .0001), and −0.84 (95% CI −1.47 to −0.22; P < .0001), respectively. Compared to active comparators, low vision rehabilitation improved vision-related QoL (SMD −0.26; 95% CI −0.46 to −0.06; P = .01) and activities of daily living (QoL: physical aspect) (SMD −0.39; 95% CI −0.67 to −0.12 P < .0001). However, no significant difference in health-related QoL and adaptation to vision loss (QoL: psychological aspect) was found between low vision rehabilitation and inactive comparators.Conclusions:This meta-analysis indicated that low vision rehabilitation interventions, particularly psychological therapies and methods of enhancing vision, may improve vision-related QoL and visual functioning in people with sight loss compared to usual care. Further studies should explore longer maintenance effects and the costs of several types of low vision rehabilitation. Studies characterizing the mechanisms of rehabilitation interventions in different settings, including low-income countries, are also required.     

Transient oculomotor paralysis after cerebral angiography: A case report

Rationale:A special case of transient oculomotor nerve palsy after cerebral angiography.Patient concerns:A 55-year-old man developed oculomotor nerve dysfunction after right radial artery puncture angiography.Diagnoses:Cerebral angiography-induced oculomotor nerve palsy.Interventions:According to the patient''s disease state, intravenous drip of dexamethasone 10 mg/d.Outcomes:Magnetic resonance imaging (MRI) showed no abnormalities, and the patient recovered completely after 48 hours of hormone therapy.Lessons:Transient eye palsy caused by contrast agent encephalopathy is a clinically rare neurological dysfunction caused by adverse effects of contrast agents. Early prevention and correct treatment are critical.     

Photodynamic therapy for radiation maculopathy: A case report

Rationale:The best treatment protocol for radiation maculopathy in children has not been determined. The purpose of this study was to determine the effect of photodynamic therapy (PDT) on radiation maculopathy.Patient concerns:An 11-year-old boy who was originally diagnosed with orbital rhabdomyosarcoma when he was 1 year old, in October 2008. The lesion improved after peripheral blood stem cell transplantation, chemotherapy and radiation therapy. A cataract was detected in his right eye in May 2011, and he underwent cataract surgery in July 2011. Continuous amblyopia training maintained his visual acuity in his right eye. In January 2017, his visual acuity was reduced and macular edema was detected with optical coherence tomography.Diagnoses:We diagnosed radiation maculopathy, from the history of radiation therapy, macular edema by optical coherence tomography, and hyperfluorescent site by fluorescein angiography.Interventions:We performed PDT in June 2017.Outcomes:Treatment with PDT improved macular edema and his visual acuity.Lessons:Radiation retinopathy is progressive disorder with poor prognosis. PDT could be considered to treat radiation maculopathy.     

Radiotherapy of granulomatosis with polyangiitis occurring in the eyelid: A case report and literature review

Introduction:Granulomatosis with polyangiitis (GPA) is a chronic systemic vasculitis characterized by necrotizing granulomatous vasculitis. The disease mainly affects the middle and small blood vessels and mainly occurs in the upper respiratory tract (nose and paranasal sinuses), lower respiratory tract (lungs), and kidneys. Disease occurrence in the eyelid area is relatively rare. The standard GPA treatment is combination therapy with adrenocortical hormone and immunosuppressants. Radiotherapy as a treatment option for GPA has not been widely investigated.Patient concerns:A 29-year-old man presented with a 1.0 × 1.0 cm mass without exophthalmos and decreased vision in the left lower eyelid. Computed tomography revealed a mass-like high-density shadow below the left eye with a computed tomography value of 80-108 U.Diagnosis:The laboratory investigations revealed positive cytoplasmic antineutrophil cytoplasmic antibodies (titer = 1:40). Biopsy of the lower left eyelid mass revealed necrosis and granulomatous reaction with a large number of inflammatory cell infiltration. After consultation with the pathology department, the diagnosis was determined as left lower eyelid GPA.Interventions:The patient received 9MeV electron beam radiation therapy in the area of the left lower eyelid lesion.Outcomes:The lesion in the patient was significantly reduced and the symptom relieved obviously. No symptom recurrence or significant toxicity occurred during or after the treatment. The patient remains under routine follow-up.Conclusion:We present a case of a male patient with GPA located exclusively in the eyelid area, who underwent successful radiotherapy and achieved a complete response. The lesson we learned from this case study is that for GPA patients, when the standard treatment model fails to achieve good results, novel treatments such as radiotherapy should be considered according to the situation.     

Subcutaneous golimumab induced and maintained clinical response in a child with a biological-experienced steroid-refractory flare of ulcerative colitis: A case report

Introduction:Golimumab is a fully human antitumor necrosis monoclonal antibody that can be administered by either subcutaneous injection or intravenous infusion. Golimumab is approved for the treatment of the adults with rheumatic diseases, and ulcerative colitis, Whereas in children, golimumab is indicated only for the treatment of active polyarticular juvenile idiopathic arthritis. We have written on the off-label use of subcutaneous golimumab, which helped to induce and maintain remission on a low-weight biologically experienced child with steroid-refractory ulcerative colitis flare.Patient concerns:A 13-year-old pancolitis Syrian boy presented with abdominal pain and six to seven times bloody diarrhea. The child had treated with mesalamine 80 mg/kg/day, azathioprine 2.5 mg/kg/day, infliximab with an induction dose of 5 mg/kg at weeks 0, 2, and 6 followed by 5 mg/kg every 8 weeks. Infliximab did not maintain remission as the patient suffered from two flares that required hospital admission, intravenous corticosteroids, and infliximab escalation. Initial tests disclosed leukocytosis, anemia, hypoalbuminemia, an elevation in C-reactive protein and fecal calprotectin. All Stool studies were negative including routine stool cultures, Clostridium difficile toxin, Escherichia coli O157:H7, Cryptosporidium, and microscopy for ova and parasites. A sigmoidoscopy revealed multiple large ulcerations and spontaneous bleeding, colon biopsies were negative for Clostridium difficile and Cytomegalovirus. Cyclosporine, tacrolimus, and adalimumab were unavailable in Syria. Child''s parents opposed colectomy as a treatment option.Diagnosis:Ulcerative colitis flare.Interventions:A subcutaneous golimumab with a loading dose of 200 mg at week 0, followed by 100 mg at week 2, then 50 mg every 4 weeks.Outcomes:The patient achieved clinical remission by week sixth and maintained the remission for the next 90 weeks. At the time of last evaluation, tests, including C-reactive protein and fecal calprotectin, were within normal limits, complete colonoscopy revealed erythema, edema, mucosal friability, loss of vascular patterns, and pseudo-polyps. The Pediatric Ulcerative Colitis Activity Index and Mayo scores were 5 and 2 points, respectively. No adverse events were documented.Conclusion:Golimumab has shown potential efficacy and safety in the treatment of ulcerative colitis in children which may indicate a significant future role for subcutaneous golimumab in pediatrics ulcerative colitis.     

Measurement of postpartum blood loss using a new two-set liquid collection bag for vaginal delivery: A prospective,randomized, case control study

Background:Postpartum hemorrhage (PPH) is a major obstetric complication, and the real-time measurement of blood loss is important in the management and treatment of PPH. We designed a new two-set liquid collection bag (TSLCB) for measuring postpartum blood loss in vaginal delivery. The aim of this study was to evaluate the effectiveness of the TSLCB in separating the blood from the amniotic fluid during vaginal delivery and in determining the accuracy of the measured postpartum blood loss.Methods:A prospective, randomized, case control study was conducted in the Women''s Hospital, Zhejiang University School of Medicine, from March 2018 to April 2018. Sixty single pregnant women with spontaneous labor at 37–41 weeks without maternal complications were randomly divided into the experimental and control groups. The TSLCB was used to evaluate separately the amount of blood and amniotic fluid. For the control group, visual estimation and traditional plastic blood-collecting consumables were used to estimate the amount of postpartum blood loss. The measured blood loss between the two groups was compared, and the association of the measured blood loss with various clinical lab indices and vital signs was investigated.Results:The TSLCB (the experimental group) improved the detection of the measured blood loss compared with visual estimation and the traditional method (the control group) (P < .05). In the experimental group, correlation analysis showed that the measured blood loss at delivery and within 24 h of delivery was significantly associated with the decreased hemoglobin level, red blood cell count, and hematocrit level of patients (r = −0.574, −0.455, −0.437; r = 0.-595, −0.368, −0.374; P < .05). In the control group, only the measured blood loss within 24 h of delivery was associated with the decreased hemoglobin level (r = −0.395, P < .05). No blood transfusion and plasma expanders were required in the treatment of PPH for both groups.Conclusions:The TSLCB can be used to accurately measure the postpartum blood loss in vaginal delivery by medical personnel.     

Retinal toxicity caused by hydroxychloroquine in patients with systemic lupus erythematosus: A case report

Rationale:Hydroxychloroquine has excellent anti-inflammatory and immunomodulatory effects as one of the antimalarial drugs. In particular, hydroxychloroquine was once widely used as a treatment for the new coronavirus pneumonia epidemic in 2020. Retinopathy caused by hydroxychloroquine is normally irreversible, but little attention has been paid to it.Patient concerns:A 38-year-old young Chinese woman was taking oral hydroxychloroquine 400 mg daily to control lupus disease activity for six years after the diagnosis of systemic lupus erythematosus (SLE). She did not have any history of eye disease and was admitted to the hospital with a sudden blurring of both eyes.Diagnoses:The diagnosis of retinal macular degeneration caused by hydroxychloroquine was made after excluding other interfering diseases based on the patient''s long-term use of hydroxychloroquine and the results of the eye examination.Interventions:The patient was discontinued from hydroxychloroquine. To control the recurrence of SLE, she was given intravenous methylprednisolone, oral tacrolimus and mycophenolate. Meanwhile, she was asked to take extra care of her eyes and to come to the hospital every three months to have her vision checked.Outcomes:The patient''s blurred vision improved one week later. Three months later, her vision examination showed no further decline (0.4 in the right eye and 0.6 in the left eye). Meanwhile, the SLE disease activity index (SLEDAI) decreased from six points to five points currently.Lessons:Retinopathy caused by hydroxychloroquine is irreversible and there is no particularly effective treatment. Discontinuation of hydroxychloroquine, better daily eye protection, and regular vision checks are the keys to preventing retinopathy. Although hydroxychloroquine causing retinal toxicity was mentioned several years ago, the rate and severity of retinal toxicity require further research. How to get more patients to take care of their eyes requires continuous and increased education by doctors.     

Clinical assessment and FGFR2 mutation analysis in a Chinese family with Crouzon syndrome: A case report

Rationale:Crouzon syndrome is an autosomal dominant genetic disorder caused by mutations in fibroblast growth factor receptor 2 (FGFR2) and one of the most common types of craniosynostosis. Here we report the detection of FGFR2 mutation and its related clinical findings in 2 patients with Crouzon syndrome from a Chinese family.Patient concerns:We report a case of a 28-year-old male patient presented with the chief complaint of gradually blurring of his eyes over the last 6 months before visiting our clinics. History revealed low visual acuity in his right eye since childhood. Physical examination showed that both the patient and his mother have the appearance of craniofacial dysostosis, mandibular prognathism, ocular proptosis, short superior lip, scoliosis, and thoracic deformity.Diagnosis:Auxiliary examinations lead to the diagnosis of Crouzon syndrome with binocular optic atrophy, myelinated retina nerve fibers, and ametropia in both eyes, and amblyopia in the right eye of the male patient. The molecular genetic analysis confirmed the diagnosis by detecting a heterozygous pathogenic mutation c.1026C > G (C342W) in exon 10 of FGFR2 in both the patient and his mother, but not in any of the unaffected family members.Interventions and outcomes:None.Lessons:Our study confirms the presence of optic nerve atrophy in patients with Crouzon syndrome carrying FGFR2 C342W mutations and indicates that MRI and funduscopy should be performed to examine the optic nerve changes for patients with Crouzon syndrome.     

Effect of rituximab dose on induction therapy in ABO-incompatible living kidney transplantation: A network meta-analysis

Background:Rituximab is an induction immunosuppressant essential for ABO-incompatible kidney transplantation (ABOi KT). However, studies on its dosing, which differs among countries and transplant centers, are lacking. Therefore, we retrospectively investigated the effectiveness of the induction dose of rituximab against patient mortality, graft failure, and adverse events.Methods:We included the studies referring to at least 2 of eligible induction doses (200 mg, 200–500 mg, or 500 mg) of rituximab during ABOi KT and relevant outcomes such as patient survival, graft failure, and bacterial and viral infections. We performed direct and indirect network meta-analyses using Bayesian models and ranked different rituximab doses using generation mixed treatment comparison. Publications were retrieved using CENTRAL, MEDLINE, EMBASE, and Science Citation Index Expanded databases from 1970 to February 2020 and analyzed. The GRADE of network meta-analysis approach specified 4 levels of certainty for a given result: high, moderate, low, and very low.Results:Among the 4256 patients from 21 trials, glomerular filtration rate, graft loss, antibody-mediated rejection, T-cell mediated rejection, fungal infection, bacterial infection, and CMV infection did not differ among ABOi groups treated with different rituximab doses. The effect on mortality was significantly higher in rituximab 200 to 500 mg, and rituximab 500 mg groups (odds ratios [OR] 3.5, 95% CrI: 1.3–9.8, and OR 3.0, 95% CrI 1.1–9.8), but not in rituximab 20 mg group (OR 0.45, 95% CrI 0.036–2.5). The incidence of BK virus was significantly lower in the rituximab 200-mg group than in the other groups.Discussion:In ABO-incompatible kidney transplantation, low-dose rituximab is more efficacious than higher doses and reduces serious infection risks. Additional randomized controlled trials might be needed to confirm these findings due to small sample size.     

Bilateral retinal pigment epithelial tears in acute central serous chorioretinopathy without bullous retinal detachment: A case report

Rationale:Several reports have described retinal pigment epithelial (RPE) tears in central serous chorioretinopathy (CSC). However, there have been no reports of spontaneously large RPE tears in acute CSC without bullous retinal detachment (RD). Herein, we report and provide sequential images of a case of bilateral spontaneous large RPE tears in patient with acute CSC without bullous RD.Patient Concerns:An 88-year-old female patient was admitted with impaired vision in both eyes, which began 10 days prior. The visual acuity was 0.4 and 0.5 in the right and left eye, respectively. She had started taking oral steroids 2 weeks prior for polymyalgia.Diagnosis:Ophthalmologic examinations, including fundus photography, optical coherence tomography, and fluorescence angiography, were performed, and she was diagnosed with steroid-induced acute CSC in both eyes with large pigment epithelial detachment (PED) of approximately 4-disc diameter. Discontinuation of steroids and follow-up ophthalmic examinations were performed. However, a spontaneous large RPE tear occurred in the right eye.Interventions:We performed follow-ups more frequently and CSC treatment such as laser photocoagulation, photodynamic therapy, and anti-vascular endothelial growth factor injections were not performed.Outcomes:Nine months later, a spontaneous large RPE tear occurred sequentially in the left eye. Her final visual acuity was 0.3 and 0.15 in the right and left eye, respectively.Lessons:Patients may spontaneously develop large RPE tears in both eyes, despite no treatment for acute CSC with non-bullous RD. Large PED and old age may affect this. Therefore, for a CSC patient with a large PED and advanced age, attention must be paid when determining treatment.     

Two years outcomes of treating full-thickness macula hole associated with idiopathic macular telangiectasia type 2 by internal limiting membrane inverted flap technique: Case reports

Rational:Macular telangiectasia (MacTel) is an uncommon ocular disorder that can lead to legal blindness. MacTel type 2 is characterized by a bilateral loss of macular transparency, the presence of white crystals on the retina, aberrant blood vessel growth, and neurodegeneration of the macula. Full-thickness macular holes (FTMHs) are a prominent cause of vision reduction in MacTel type 2, and the standard care for an FTMH is pars plana vitrectomy (PPV) to restore the FTMH and best-corrected visual acuity (BCVA). However, surgical outcomes in previous reports were not good, with a lack of closure or a reopening of the FTMH, compared with those with an idiopathic FTMH. Thus, this study aimed to determine the surgical outcomes of PPV with the inverted ILM flap technique for the treatment of FTMHs with a 2-year postoperative follow-up in three patients with MacTel type 2.Patient concerns:This study involved 3 patients who had been diagnosed with MacTel type 2 at a local eye clinic and who was subsequently referred to our department for a more detailed examination.Diagnoses:Three patients were diagnosed with MacTel type 2 using dilated ophthalmoscopy, fluorescein angiography, and optical coherence tomography (OCT) in both eyes. A FTMH was developed and visual acuity decreased during follow-up period in all of the patients.Interventions:Each patient underwent PPV in 1 eye using the inverted ILM-flap technique, gas tamponade, and prone positioning.Outcomes:The FTMH was successfully closed in the 3 cases after the surgery. OCT showed that the FTMH remained closed at the last follow-up examination in 2 patients and vision improved to 20/20 and 20/25. In the other patient, the hole was closed temporarily after surgery, but was reopened at 6 months. The vision had improved to 20/60 until the hole was reopened, and it was 20/100 at the final follow-up examination.Lessons:Although only 3 patients were examined, the inverted ILM-flap technique may be an effective and safe method to close an FTMH in patients with MacTel type 2. However, the surgery cannot prevent the reopening of the hole when the retinal atrophy progresses.     

Sugammadex induced bradycardia and hypotension: A case report and literature review

Rationale:There is evidence that sugammadex can facilitate extubation post-surgery and attenuate postoperative pulmonary complications resulting from postoperative residual neuromuscular blockade. However, it may induce adverse effects, including bronchospasm, laryngospasm, bradycardia, hypotension, and cardiac arrest. Here, we present a case of sugammadex-induced bradycardia and hypotension.Patient concerns:An 82-year-old female received video-assisted thoracic surgery decortication and wedge resection of the lung for empyema. Post-surgery, she developed bradycardia, hypotension, hypoxia, and weakness.Diagnoses:The patient was suspected to have sugammadex-induced bradycardia, hypotension, hypoxia and weakness.Interventions:The patient received immediate treatment with atropine (0.5 mg) for bradycardia. Glycopyrrolate (0.1 mg) and neostigmine (1 mg) were administered to improve the train-of-four (TOF) ratio.Outcomes:Following initial management, we observed improvement in the hemodynamics of the patient. She was discharged without any sequelae.Lessons:Sugammadex-induced bradycardia or cardiac arrest are rare; however, anesthesiologists must consider the possibility of the occurrence of such events and initiate appropriate management measures. Immediate treatment with atropine and inotropic or vasopressors is warranted if the patient presents with bradycardia.