Acta Ophthalmologica 2015
(A)
(B)
(C)
(E)
(G)
(D)
(F)
(H)
and subretinal neovascularization. Nihon Ganka Gakkai Zasshi 100: 705–709. Kindler P (1970): Morning glory syndrome: unusual congenital optic disk anomaly. Am J Ophthalmol 69: 376–384. Sobol WM, Bratton AR, Rivers MB et al. (1990): Morning glory disk syndrome associated with subretinalneovascular membrane formation. Am J Ophthalmol 110: 93–94.
(I)
(J)
Fig. 1. Baseline evaluation of the left eye. (A) Multicolour image showing morning glory syndrome with glial tissue overlying the optic disc cup and subretinal haemorrhage. (B) Fluorescein angiography revealed late-stage peripapillary neovascularization. (C) Indocyanine green angiography revealed hyperfluorescence of the lesion. (D) B-scan echography showed conical excavation of the posterior pole (arrow). (E) Spectral-domain optical coherence tomography (SD-OCT) showed subretinal tissue proliferation with subretinal fluid in the peripapillary area. (F) SD-OCT showed an abnormal communication between the subarachnoid space and vitreous space. (G–I) Multicolour image, fluorescein angiography and indocyanine green angiography, respectively, showing total regression of the CNV after four months of treatment. (J) SDOCT revealed complete resolution of the subretinal fluid.
have been described (Sobol et al. 1990; Chuman et al. 1996), and in only one of these cases was it adjacent to the optic disc. This case report suggests that CNV in eyes affected by MGS may result from hemodynamic or mechanical changes at the border of the staphyloma as occurs in tilted disc syndrome (Arias & Mones 2010).
References Arias L & Mones J (2010): Ranibizumab in the treatment of choroidal neovascularization on the border of an inferior staphyloma associated with tilted disc syndrome. Clin Ophthalmol 4: 227–231. Cennamo G, de Crecchio G, Iaccarino G et al. (2010): Evaluation of morning glory syndrome with spectral optical coherence tomography and echography. Ophthalmology 117: 1269–1273. Chuman H, Nao-I N & Sawada A (1996): A case of morning glory syndrome associated with contractile movement of the optic disc
Correspondence: Gilda Cennamo, MD Eye Clinic Department of Neurosciences, Reproductive Sciences and Dentistry University of Naples Federico II Via Pansini 5, Naples 80100 Italy Tel: +39 0817463731 Fax: +39 0817462383 Email: [email protected]
First report of Candida palmioleophila endogenous endophthalmitis Nigel Datta,1 Maiken Cavling Arendrup2 and Jon Peiter Saunte3 1
Department of Ophthalmology, Copenhagen University Hospital Roskilde, Roskilde, Denmark; 2Unit of Mycology, Statens Serum Institut, Copenhagen, Denmark; 3Department of Ophthalmology, Copenhagen University Hospital Glostrup, Glostrup, Denmark doi: 10.1111/aos.12662
Editor, ndogenous fungal endophthalmitis (EFE) by Candida palmioleophila (Cp) has to our knowledge not previously been reported. Ocular candidiasis complicates candidaemia at a rate of approximately 2–26% (Ullmann et al. 2012). The majority of cases are chorioretinitis whilst EFE is less common (Ullmann et al. 2012). Symptoms of EFE are reduced vision, pain and photophobia, and findings are conjunctival hyperaemia, infiltrates in the choroid, retina or vitreous (Osthoff et al. 2006). Cp can be misdiagnosed as C. famata or C. guilliermondii (Sugita et al. 1999; Jensen & Arendrup 2011). In contrast to those species, Cp is susceptible to echinocandins but highly fluconazole resistant.
E
A 51-year-old man with acute myeloid leukaemia was admitted with acute Pneumococcus pneumonia. The patient complained of blurred vision and floaters in the left eye (LE). Ophthalmoscopy revealed multiple preretinal haemorrhages progressing to LE vitritis and multiple new preretinal haemorrhages and creamy white infiltrates bilaterally 1 week later (Fig. 1). Peripheral blood cultures yielded Candida initially identified as C. guilliermondii. Intravenous fluconazole (200 mg BID) was initiated. Susceptibility pattern (Etest) was as follows: amphotericin B, voriconazole, caspofungin susceptible and fluconazole intermediate. At the reference laboratory, the isolate was re-identified as Cp with the following EUCAST MICs (mg/L): fluconazole MIC>16 (R), itraconazole 0.125 (S), voriconazole 0.25 (S according to the CLSI breakpoints, R according to the EUCAST breakpoints), posaconazole 0.03 (S), caspofungin 1 (S) and Etest MIC: amphotericin B 0.047 (S). Due to insufficient clinical effect and the new susceptibility results, treatment was 4 weeks later changed to amphotericin B (50 mg QD) for 3 weeks, then combined with 5flucytosine (2500 mg QID) intravenously for 3 weeks and finally changed to oral voriconazole (200 mg BID) for 4 weeks. Increased inflammation and white choroidal infiltrates developed in both eyes. Vitrectomy with intravitreal injection of amphotericin B was performed in both eyes, which resulted in gradual improvement (Fig. 1). A vitreous sample on day 58 was microscopy and culture negative. At 3-year follow-up, there was no sign of recurrence, and visual acuity was 6/6 in RE and 6/9 in LE. Cp is intrinsically highly resistant to fluconazole and shows moderately reduced susceptibility to voriconazole, which may compromise efficacy unless high exposure can be achieved (Jensen & Arendrup 2011). Although Cp is echinocandin susceptible, echinocandins are less attractive for EFE due to the poor penetration across the blood–brain barrier. Therefore, initial combination therapy of intravitrous amphotericine B, intravenous flucytosine and intravitreal installation of amphotericin B deoxycholate potentially followed by voriconazole if therapeutic drug monitoring ensures significant drug levels currently appears as the optimal treatment of EFE by Cp (Barza 1998). Optimal and timely management of EFE necessitate a close collaboration
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Acta Ophthalmologica 2015
Fig. 1. Fundus photography mosaics of right eye at day 0 (left) and at day 28 (right). Left: Preretinal haemorrhages and creamy white infiltrates. Right: Reduced haemorrhages and gradually decreasing infiltrates and development of scar tissue.
between a laboratory with mycological expertise and a skilled ophthalmologist (Jensen & Arendrup 2011).
Isolated oculomotor nerve palsy revealing infectious mononucleosis
References
Ariane Malcles, Solene Ronsin, Emilie Agard, Lucie Abouaf, Caroline Tilikete, Alain Vighetto and Damien Biotti
Barza M (1998): Editorial response: treatment options for Candidal endophthalmitis. Clin Infect Dis 27: 1134–1136. Jensen RH & Arendrup MC (2011): Candida palmioleophila: characterization of a previously overlooked pathogen and its unique susceptibility profile in comparison with five related species. J Clin Microbiol 49: 549–556. Osthoff M, Hilge R, Schulze-D€ obold C & Bogner JR (2006): Endogenous endophthalmitis with azole-resistant Candida albicans-case report and review of the literature. Infection 34: 85–88. Sugita T, Kagaya K, Takashima M et al. (1999): A clinical isolate of Candida palmioleophila formerly identified as Torulopsis Candida. Nihon Ishinkin Gakkai Zasshi 40: 21–25. Ullmann AJ, Cornely OA, Donnelly JP et al. (2012): ESCMID guideline for the diagnosis and management of Candida diseases. developing European guidelines in clinical microbiology and infectious diseases. Clin Microbiol Infect 18(Suppl. 7): 1–8.
Correspondence: Nigel Datta Department of Ophthalmology Copenhagen University Hospital Roskilde Koegevej 7-13 4000 Roskilde Denmark Tel: +45 47323900 Fax: +45 46362645 Email: [email protected]
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Neuro-Ophthalmology Unit, Lyon Civil Hospitals, Neurological Hospital, Lyon 1 University, Bron Cedex, France doi: 10.1111/aos.12636
Editor, pstein–Barr virus (EBV) is a member of the herpesvirus family causing infectious mononucleosis (IM). Neurological complications have been reported rarely. A previously healthy 18-year-old woman presented with a 5-day history of binocular vertical diplopia, asthenia and night sweats. Ophthalmological examination was normal. Pupils were equal and reactive to light, with no relative afferent pupillary defect. Oculomotor examination revealed a left pupil sparing partial third nerve palsy (partial ptosis and moderate restriction of elevation, depression and adduction). The rest of the neurological examination was unremarkable. General examination revealed an apyretic patient with painless disseminated lymph nodes and a mild hepatomegaly. Brain MRI showed a focal T2 abnormal hyperintense signal located on the left oculomotor nerve at its exit from the mesencephalon. A gadolinium enhancement was noted (Fig. 1).
E
Biochemistry abnormalities showed hepatic cytolysis (ASAT/ALAT 3ULN) and C-reactive protein level of 13.5 mg/ l. IgM and IgG antibodies against Epstein–Barr viral capsid antigen were found positive, whereas IgG antibodies for Epstein–Barr nuclear antigen were negative. Epstein–Barr viral load testing by PCR reached 65 000 copies/ml. This biological profile was indicative of an acute primary EBV infection. Diplopia gradually resolved in a few days, followed in several weeks by the resolution of asthenia and lymphadenopathy. At the 2-month follow-up visit, neuro-ophthalmological examination and liver enzymes level had return to normal. A new brain MRI showed the decrease in size of the previously noted hyperintense signal of the left oculomotor nerve, with mild residual gadolinium enhancement. Peripheral and/or central nervous system (CNS) involvement is rare in IM, occurring in
(A)
(B)
(C)
(E)
(G)
(D)
(F)
(H)
and subretinal neovascularization. Nihon Ganka Gakkai Zasshi 100: 705–709. Kindler P (1970): Morning glory syndrome: unusual congenital optic disk anomaly. Am J Ophthalmol 69: 376–384. Sobol WM, Bratton AR, Rivers MB et al. (1990): Morning glory disk syndrome associated with subretinalneovascular membrane formation. Am J Ophthalmol 110: 93–94.
(I)
(J)
Fig. 1. Baseline evaluation of the left eye. (A) Multicolour image showing morning glory syndrome with glial tissue overlying the optic disc cup and subretinal haemorrhage. (B) Fluorescein angiography revealed late-stage peripapillary neovascularization. (C) Indocyanine green angiography revealed hyperfluorescence of the lesion. (D) B-scan echography showed conical excavation of the posterior pole (arrow). (E) Spectral-domain optical coherence tomography (SD-OCT) showed subretinal tissue proliferation with subretinal fluid in the peripapillary area. (F) SD-OCT showed an abnormal communication between the subarachnoid space and vitreous space. (G–I) Multicolour image, fluorescein angiography and indocyanine green angiography, respectively, showing total regression of the CNV after four months of treatment. (J) SDOCT revealed complete resolution of the subretinal fluid.
have been described (Sobol et al. 1990; Chuman et al. 1996), and in only one of these cases was it adjacent to the optic disc. This case report suggests that CNV in eyes affected by MGS may result from hemodynamic or mechanical changes at the border of the staphyloma as occurs in tilted disc syndrome (Arias & Mones 2010).
References Arias L & Mones J (2010): Ranibizumab in the treatment of choroidal neovascularization on the border of an inferior staphyloma associated with tilted disc syndrome. Clin Ophthalmol 4: 227–231. Cennamo G, de Crecchio G, Iaccarino G et al. (2010): Evaluation of morning glory syndrome with spectral optical coherence tomography and echography. Ophthalmology 117: 1269–1273. Chuman H, Nao-I N & Sawada A (1996): A case of morning glory syndrome associated with contractile movement of the optic disc
Correspondence: Gilda Cennamo, MD Eye Clinic Department of Neurosciences, Reproductive Sciences and Dentistry University of Naples Federico II Via Pansini 5, Naples 80100 Italy Tel: +39 0817463731 Fax: +39 0817462383 Email: [email protected]
First report of Candida palmioleophila endogenous endophthalmitis Nigel Datta,1 Maiken Cavling Arendrup2 and Jon Peiter Saunte3 1
Department of Ophthalmology, Copenhagen University Hospital Roskilde, Roskilde, Denmark; 2Unit of Mycology, Statens Serum Institut, Copenhagen, Denmark; 3Department of Ophthalmology, Copenhagen University Hospital Glostrup, Glostrup, Denmark doi: 10.1111/aos.12662
Editor, ndogenous fungal endophthalmitis (EFE) by Candida palmioleophila (Cp) has to our knowledge not previously been reported. Ocular candidiasis complicates candidaemia at a rate of approximately 2–26% (Ullmann et al. 2012). The majority of cases are chorioretinitis whilst EFE is less common (Ullmann et al. 2012). Symptoms of EFE are reduced vision, pain and photophobia, and findings are conjunctival hyperaemia, infiltrates in the choroid, retina or vitreous (Osthoff et al. 2006). Cp can be misdiagnosed as C. famata or C. guilliermondii (Sugita et al. 1999; Jensen & Arendrup 2011). In contrast to those species, Cp is susceptible to echinocandins but highly fluconazole resistant.
E
A 51-year-old man with acute myeloid leukaemia was admitted with acute Pneumococcus pneumonia. The patient complained of blurred vision and floaters in the left eye (LE). Ophthalmoscopy revealed multiple preretinal haemorrhages progressing to LE vitritis and multiple new preretinal haemorrhages and creamy white infiltrates bilaterally 1 week later (Fig. 1). Peripheral blood cultures yielded Candida initially identified as C. guilliermondii. Intravenous fluconazole (200 mg BID) was initiated. Susceptibility pattern (Etest) was as follows: amphotericin B, voriconazole, caspofungin susceptible and fluconazole intermediate. At the reference laboratory, the isolate was re-identified as Cp with the following EUCAST MICs (mg/L): fluconazole MIC>16 (R), itraconazole 0.125 (S), voriconazole 0.25 (S according to the CLSI breakpoints, R according to the EUCAST breakpoints), posaconazole 0.03 (S), caspofungin 1 (S) and Etest MIC: amphotericin B 0.047 (S). Due to insufficient clinical effect and the new susceptibility results, treatment was 4 weeks later changed to amphotericin B (50 mg QD) for 3 weeks, then combined with 5flucytosine (2500 mg QID) intravenously for 3 weeks and finally changed to oral voriconazole (200 mg BID) for 4 weeks. Increased inflammation and white choroidal infiltrates developed in both eyes. Vitrectomy with intravitreal injection of amphotericin B was performed in both eyes, which resulted in gradual improvement (Fig. 1). A vitreous sample on day 58 was microscopy and culture negative. At 3-year follow-up, there was no sign of recurrence, and visual acuity was 6/6 in RE and 6/9 in LE. Cp is intrinsically highly resistant to fluconazole and shows moderately reduced susceptibility to voriconazole, which may compromise efficacy unless high exposure can be achieved (Jensen & Arendrup 2011). Although Cp is echinocandin susceptible, echinocandins are less attractive for EFE due to the poor penetration across the blood–brain barrier. Therefore, initial combination therapy of intravitrous amphotericine B, intravenous flucytosine and intravitreal installation of amphotericin B deoxycholate potentially followed by voriconazole if therapeutic drug monitoring ensures significant drug levels currently appears as the optimal treatment of EFE by Cp (Barza 1998). Optimal and timely management of EFE necessitate a close collaboration
e517
Acta Ophthalmologica 2015
Fig. 1. Fundus photography mosaics of right eye at day 0 (left) and at day 28 (right). Left: Preretinal haemorrhages and creamy white infiltrates. Right: Reduced haemorrhages and gradually decreasing infiltrates and development of scar tissue.
between a laboratory with mycological expertise and a skilled ophthalmologist (Jensen & Arendrup 2011).
Isolated oculomotor nerve palsy revealing infectious mononucleosis
References
Ariane Malcles, Solene Ronsin, Emilie Agard, Lucie Abouaf, Caroline Tilikete, Alain Vighetto and Damien Biotti
Barza M (1998): Editorial response: treatment options for Candidal endophthalmitis. Clin Infect Dis 27: 1134–1136. Jensen RH & Arendrup MC (2011): Candida palmioleophila: characterization of a previously overlooked pathogen and its unique susceptibility profile in comparison with five related species. J Clin Microbiol 49: 549–556. Osthoff M, Hilge R, Schulze-D€ obold C & Bogner JR (2006): Endogenous endophthalmitis with azole-resistant Candida albicans-case report and review of the literature. Infection 34: 85–88. Sugita T, Kagaya K, Takashima M et al. (1999): A clinical isolate of Candida palmioleophila formerly identified as Torulopsis Candida. Nihon Ishinkin Gakkai Zasshi 40: 21–25. Ullmann AJ, Cornely OA, Donnelly JP et al. (2012): ESCMID guideline for the diagnosis and management of Candida diseases. developing European guidelines in clinical microbiology and infectious diseases. Clin Microbiol Infect 18(Suppl. 7): 1–8.
Correspondence: Nigel Datta Department of Ophthalmology Copenhagen University Hospital Roskilde Koegevej 7-13 4000 Roskilde Denmark Tel: +45 47323900 Fax: +45 46362645 Email: [email protected]
e518
Neuro-Ophthalmology Unit, Lyon Civil Hospitals, Neurological Hospital, Lyon 1 University, Bron Cedex, France doi: 10.1111/aos.12636
Editor, pstein–Barr virus (EBV) is a member of the herpesvirus family causing infectious mononucleosis (IM). Neurological complications have been reported rarely. A previously healthy 18-year-old woman presented with a 5-day history of binocular vertical diplopia, asthenia and night sweats. Ophthalmological examination was normal. Pupils were equal and reactive to light, with no relative afferent pupillary defect. Oculomotor examination revealed a left pupil sparing partial third nerve palsy (partial ptosis and moderate restriction of elevation, depression and adduction). The rest of the neurological examination was unremarkable. General examination revealed an apyretic patient with painless disseminated lymph nodes and a mild hepatomegaly. Brain MRI showed a focal T2 abnormal hyperintense signal located on the left oculomotor nerve at its exit from the mesencephalon. A gadolinium enhancement was noted (Fig. 1).
E
Biochemistry abnormalities showed hepatic cytolysis (ASAT/ALAT 3ULN) and C-reactive protein level of 13.5 mg/ l. IgM and IgG antibodies against Epstein–Barr viral capsid antigen were found positive, whereas IgG antibodies for Epstein–Barr nuclear antigen were negative. Epstein–Barr viral load testing by PCR reached 65 000 copies/ml. This biological profile was indicative of an acute primary EBV infection. Diplopia gradually resolved in a few days, followed in several weeks by the resolution of asthenia and lymphadenopathy. At the 2-month follow-up visit, neuro-ophthalmological examination and liver enzymes level had return to normal. A new brain MRI showed the decrease in size of the previously noted hyperintense signal of the left oculomotor nerve, with mild residual gadolinium enhancement. Peripheral and/or central nervous system (CNS) involvement is rare in IM, occurring in
