Case Report
Full-thickness macular hole in Bartonella henselae neuroretinitis in an 11-year-old girl Anisha Seth, Usha K. Raina, Sriram Thirumalai, Supriya Batta, Basudeb Ghosh Department of Ophthalmology, Guru Nanak Eye Centre, New Delhi, India
Cat scratch disease is a febrile illness caused by Bartonella henselae and is associated with rash at the site of cat bite or scratch and regional lymphadenopathy. Various ocular manifestations of cat scratch disease have been described, mainly retinochoroiditis, optic disc swelling, neuroretinitis, vascular occlusive events, serous retinal detachment, vitreous hemorrhage, intermediate uveitis, inflammatory lesions of the optic
Introduction
nerve head and rarely full thickness macular hole. We describe a case of an 11-year-old girl who presented 2 weeks after the onset of symptoms, with B. Henselae neuroretinitis with full thickness macular hole at presentation. Keywords: Cat scratch disease, full thickness macular hole, neuroretinitis
described with neuroretinitis and full thickness macular hole at presentation with a short history of 2 weeks.
Cat scratch disease is caused by facultative intracellular Gram-negative bacteria Bartonella henselae. Regional lymphadenopathy (axillary, head and neck, inguinal) is the predominant clinical feature of cat scratch disease.[1] Rarely, hematogenous spread of the initial infection may result in ocular and neurological complications.[2] Various ocular manifestations of cat scratch disease have been described, that include retinochoroiditis, optic disc swelling, neuroretinitis, vascular occlusive events, serous retinal detachment, vitreous hemorrhage, intermediate uveitis and inflammatory lesions of the optic nerve head.[3-5] Three cases of full thickness macular hole in neuroretinitis have been described.[6-8] However, in all the described cases, macular hole was seen at least 1 month after the onset of symptoms. We describe a case of an 11-year-old girl who presented 2 weeks after the onset of symptoms, with B. henselae neuroretinitis along with a full thickness macular hole at presentation. To the best of our knowledge, no case has been Access this article online Quick Response Code: Website: www.ojoonline.org DOI: 10.4103/0974-620X.149866
Case Report An 11-year-old girl presented to our hospital with a complaint of decrease of vision in right eye for 2 weeks. She had a history of low-grade fever 3 weeks back. There was no history suggestive of upper respiratory tract infection, gastrointestinal infection or urinary tract infection. The patient had a history of keeping pet cats and of scratches by the same. Best-corrected visual acuity (BCVA) in the right eye was counting fingers at one foot and in the left eye was 20/20. A relative afferent pupillary defect was seen in the right eye. There were no cells or flare in the anterior chamber or vitreous cavity in either eye. Fundus examination in the right eye showed a diffusely hyperemic optic disc with surrounding white chorioretinal lesions. There was also a full thickness macular hole with surrounding serous macular detachment and hard exudates at the macula in a stellate pattern. The veins were dilated and mildly tortuous. Few hard exudates were present in the nasal retina, about 4 disc diameters away from the disc. The rest of the retina was unremarkable [Figure 1]. The left eye fundus was within normal limits. Spectral domain optical coherence tomography (SD-OCT) of the right eye showed a full thickness macular hole with neurosensory detachment and subretinal fluid. There was also an epiretinal membrane that was adherent to the retina [Figure 2].
Copyright: © 2015 Seth A, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Correspondence: Dr. Anisha Seth, Bn-56, Shalimar Bagh East, New Delhi - 110 088, India. E-mail: [email protected]
44
Oman Journal of Ophthalmology, Vol. 8, No. 1, 2015
Seth, et al.: Macular hole in neuroretinitis in an 11-year-old girl Magnetic resonance imaging of the brain and orbits was normal. The hemogram of the patient was within normal limits. However, both erythrocyte sedimentation rate and C-reactive protein were elevated at 76 mm/h and 8 mg/dL, respectively. The antibody titers against the TORCH (Toxoplasma, Rubella, Coxsackie, Herpes) group of infections were within normal limits. Chest X-ray and tuberculin skin test were also normal. Both the IgM titers and IgG titers against B. Henselae were elevated at 1:32 and 1:1024. The patient was given a course of oral prednisolone at 1 mg/kg/day for 3 weeks that was tapered off over the next 3 weeks. As the patient was immuncompetent, and did not have any systemic features like fever and lymphadenitis at presentation, no antibiotics were administered to her. At the end of 6 weeks, the BCVA in the right eye improved to 20/40. The fundus in the right eye showed resolution of optic disc edema and chorioretinal lesions with residual disc pallor. However, there was an increase in the size of macular hole with persistence of subretinal fluid [Figure 3]. SD-OCT of the macula showed an increase in the macular hole size with persistence of epiretinal membrane [Figure 4]. Surgery for macular hole was not considered as the residual visual loss was felt to be predominantly from optic neuropathy.
Discussion Cat scratch disease is typically a benign and self-limited illness lasting 6-12 weeks in the absence of antibiotic therapy. Between 25% and 60% of patients report a primary cutaneous inoculation lesion (0.5–1 cm papule or pustule) at the site of a cat scratch or bite.[1] Ocular involvement of cat-scratch disease occurs in 5–10% of cases and is the most common non-lymphatic organ involvement. It was first described by Henri Parinaud when he reported three patients with chronic fever, regional lymphadenopathy, and follicular conjunctivitis. This form of ocular involvement, known as Parinaud’s oculoglandular syndrome, is the most common ocular manifestation of cat scratch disease and occurs in 5% of cases. The most common posterior segment finding in cat-scratch disease is retinochoroiditis followed by optic disc swelling.[4] Other posterior segment manifestations are vascular occlusive events, serous retinal detachment, vitreous hemorrhage, intermediate uveitis and inflammatory lesions of the optic nerve head.[3-5] Neuroretinitis was seen in 1–2% of systemic cat scratch disease and is associated with a mild-to-moderate degree of inflammation, optic disc swelling, and delayed macular star formation. It is, usually, unilateral although bilateral involvement has been described.[9] Macular hole following inflammatory diseases has been reported in idiopathic posterior uveitis, presumed ocular histoplasmosis syndrome, fungal endophthalmitis, intraocular nasal T/natural killer lymphoma, immune recovery uveitis, and juvenile
Figure 1: Right eye fundus showing hyperemic optic disc (blue arrow) with surrounding chorioretinal lesions (yellow arrow), full thickness macular hole with serous macular detachment (red arrow) and hard exudates at macula in stellate pattern
Figure 2: Spectral domain optical coherence tomography of right macula showing full thickness macular hole with subretinal fluid with epiretinal membrane (red arrow)
Figure 3: Fundus of the right eye 6 weeks after treatment with oral prednisolone showing disc pallor (blue arrow) and a larger macular hole with subretinal fluid (red arrow)
Oman Journal of Ophthalmology, Vol. 8, No. 1, 2015
Figure 4: Spectral domain optical coherence tomography of the right macula at 6 weeks showing an increase in size of macular hole with persistent epiretinal membrane (red arrow)
45
Seth, et al.: Macular hole in neuroretinitis in an 11-year-old girl rheumatoid arthritis after cataract extraction.[6,10] Three cases of full thickness macular hole have also been described in B. Henselae neuroretinitis.[6-8] The pathogenesis of macular hole in inflammatory diseases is not well-understood. It has been hypothesized that the contraction of premacular vitreous due to inflammation causes tangential traction causing a full-thickness macular hole.[6,10] A lamellar macular hole has also been seen to develop after cystoid macular edema in inflammatory diseases. Three cases have been described where patients presented with neuroretinitis and developed macular hole over a follow up of 1-2 months. To the best of our knowledge, no case has been reported with neuroretinitis and full thickness macular hole at presentation, with a history as short as 2 weeks. The macular hole in our patient was also seen to increase in size over the follow up. This probably represents a progressive vitreous traction and the inflammatory membrane contraction despite control of disease and visual improvement. Hence, all cases with neuroretinitis should be evaluated and followed up for development of full-thickness macular hole.
References 1.
46
Carithers HA. Cat-scratch disease. An overview based on a study of 1,200 patients. Am J Dis Child 1985;139:1124-33.
2. 3.
4.
5.
6. 7. 8.
9.
10.
Ormerod LD, Dailey JP. Ocular manifestations of cat-scratch disease. Curr Opin Ophthalmol 1999;10:209-16. Ormerod LD, Skolnick KA, Menosky MM, Pavan PR, Pon DM. Retinal and choroidal manifestations of cat-scratch disease. Ophthalmology 1998;105:1024-31. Solley WA, Martin DF, Newman NJ, King R, Callanan DG, Zacchei T, et al. Cat scratch disease: Posterior segment manifestations. Ophthalmology 1999;106:1546-53. Cunningham ET Jr, McDonald HR, Schatz H, Johnson RN, Ai E, Grand MG. Inflammatory mass of the optic nerve head associated with systemic Bartonella henselae infection. Arch Ophthalmol 1997;115:1596-7. Albini TA, Lakhanpal RR, Foroozan R, Holz ER. Macular hole in cat scratch disease. Am J Ophthalmol 2005;140:149-51. Donnio A, Jean-Charles A, Merle H. Macular hole following Bartonella henselae neuroretinitis. Eur J Ophthalmol 2008;18:456-8. Alterman MA, Young BK, Eggenberger ER, Kaufman DI. Macular hole: A rare complication of ocular bartonellosis. J Neuroophthalmol 2013;33:153-4. Metz CH, Buer J, Bornfeld N, Lipski A. Bilateral Bartonella henselae neuroretinitis with stellate maculopathy in a 6-year-old boy. Infection 2012;40:191-4. Nussenblatt RB. Macular alterations secondary to intraocular inflammatory disease. Ophthalmology 1986;93:984-8.
Cite this article as: Seth A, Raina UK, Thirumalai S, Batta S, Ghosh B. Fullthickness macular hole in Bartonella henselae neuroretinitis in an 11-year-old girl. Oman J Ophthalmol 2015;8:44-6. Source of Support: Nil, Conflict of Interest: None declared.
Oman Journal of Ophthalmology, Vol. 8, No. 1, 2015
Copyright of Oman Journal of Ophthalmology is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Full-thickness macular hole in Bartonella henselae neuroretinitis in an 11-year-old girl Anisha Seth, Usha K. Raina, Sriram Thirumalai, Supriya Batta, Basudeb Ghosh Department of Ophthalmology, Guru Nanak Eye Centre, New Delhi, India
Cat scratch disease is a febrile illness caused by Bartonella henselae and is associated with rash at the site of cat bite or scratch and regional lymphadenopathy. Various ocular manifestations of cat scratch disease have been described, mainly retinochoroiditis, optic disc swelling, neuroretinitis, vascular occlusive events, serous retinal detachment, vitreous hemorrhage, intermediate uveitis, inflammatory lesions of the optic
Introduction
nerve head and rarely full thickness macular hole. We describe a case of an 11-year-old girl who presented 2 weeks after the onset of symptoms, with B. Henselae neuroretinitis with full thickness macular hole at presentation. Keywords: Cat scratch disease, full thickness macular hole, neuroretinitis
described with neuroretinitis and full thickness macular hole at presentation with a short history of 2 weeks.
Cat scratch disease is caused by facultative intracellular Gram-negative bacteria Bartonella henselae. Regional lymphadenopathy (axillary, head and neck, inguinal) is the predominant clinical feature of cat scratch disease.[1] Rarely, hematogenous spread of the initial infection may result in ocular and neurological complications.[2] Various ocular manifestations of cat scratch disease have been described, that include retinochoroiditis, optic disc swelling, neuroretinitis, vascular occlusive events, serous retinal detachment, vitreous hemorrhage, intermediate uveitis and inflammatory lesions of the optic nerve head.[3-5] Three cases of full thickness macular hole in neuroretinitis have been described.[6-8] However, in all the described cases, macular hole was seen at least 1 month after the onset of symptoms. We describe a case of an 11-year-old girl who presented 2 weeks after the onset of symptoms, with B. henselae neuroretinitis along with a full thickness macular hole at presentation. To the best of our knowledge, no case has been Access this article online Quick Response Code: Website: www.ojoonline.org DOI: 10.4103/0974-620X.149866
Case Report An 11-year-old girl presented to our hospital with a complaint of decrease of vision in right eye for 2 weeks. She had a history of low-grade fever 3 weeks back. There was no history suggestive of upper respiratory tract infection, gastrointestinal infection or urinary tract infection. The patient had a history of keeping pet cats and of scratches by the same. Best-corrected visual acuity (BCVA) in the right eye was counting fingers at one foot and in the left eye was 20/20. A relative afferent pupillary defect was seen in the right eye. There were no cells or flare in the anterior chamber or vitreous cavity in either eye. Fundus examination in the right eye showed a diffusely hyperemic optic disc with surrounding white chorioretinal lesions. There was also a full thickness macular hole with surrounding serous macular detachment and hard exudates at the macula in a stellate pattern. The veins were dilated and mildly tortuous. Few hard exudates were present in the nasal retina, about 4 disc diameters away from the disc. The rest of the retina was unremarkable [Figure 1]. The left eye fundus was within normal limits. Spectral domain optical coherence tomography (SD-OCT) of the right eye showed a full thickness macular hole with neurosensory detachment and subretinal fluid. There was also an epiretinal membrane that was adherent to the retina [Figure 2].
Copyright: © 2015 Seth A, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Correspondence: Dr. Anisha Seth, Bn-56, Shalimar Bagh East, New Delhi - 110 088, India. E-mail: [email protected]
44
Oman Journal of Ophthalmology, Vol. 8, No. 1, 2015
Seth, et al.: Macular hole in neuroretinitis in an 11-year-old girl Magnetic resonance imaging of the brain and orbits was normal. The hemogram of the patient was within normal limits. However, both erythrocyte sedimentation rate and C-reactive protein were elevated at 76 mm/h and 8 mg/dL, respectively. The antibody titers against the TORCH (Toxoplasma, Rubella, Coxsackie, Herpes) group of infections were within normal limits. Chest X-ray and tuberculin skin test were also normal. Both the IgM titers and IgG titers against B. Henselae were elevated at 1:32 and 1:1024. The patient was given a course of oral prednisolone at 1 mg/kg/day for 3 weeks that was tapered off over the next 3 weeks. As the patient was immuncompetent, and did not have any systemic features like fever and lymphadenitis at presentation, no antibiotics were administered to her. At the end of 6 weeks, the BCVA in the right eye improved to 20/40. The fundus in the right eye showed resolution of optic disc edema and chorioretinal lesions with residual disc pallor. However, there was an increase in the size of macular hole with persistence of subretinal fluid [Figure 3]. SD-OCT of the macula showed an increase in the macular hole size with persistence of epiretinal membrane [Figure 4]. Surgery for macular hole was not considered as the residual visual loss was felt to be predominantly from optic neuropathy.
Discussion Cat scratch disease is typically a benign and self-limited illness lasting 6-12 weeks in the absence of antibiotic therapy. Between 25% and 60% of patients report a primary cutaneous inoculation lesion (0.5–1 cm papule or pustule) at the site of a cat scratch or bite.[1] Ocular involvement of cat-scratch disease occurs in 5–10% of cases and is the most common non-lymphatic organ involvement. It was first described by Henri Parinaud when he reported three patients with chronic fever, regional lymphadenopathy, and follicular conjunctivitis. This form of ocular involvement, known as Parinaud’s oculoglandular syndrome, is the most common ocular manifestation of cat scratch disease and occurs in 5% of cases. The most common posterior segment finding in cat-scratch disease is retinochoroiditis followed by optic disc swelling.[4] Other posterior segment manifestations are vascular occlusive events, serous retinal detachment, vitreous hemorrhage, intermediate uveitis and inflammatory lesions of the optic nerve head.[3-5] Neuroretinitis was seen in 1–2% of systemic cat scratch disease and is associated with a mild-to-moderate degree of inflammation, optic disc swelling, and delayed macular star formation. It is, usually, unilateral although bilateral involvement has been described.[9] Macular hole following inflammatory diseases has been reported in idiopathic posterior uveitis, presumed ocular histoplasmosis syndrome, fungal endophthalmitis, intraocular nasal T/natural killer lymphoma, immune recovery uveitis, and juvenile
Figure 1: Right eye fundus showing hyperemic optic disc (blue arrow) with surrounding chorioretinal lesions (yellow arrow), full thickness macular hole with serous macular detachment (red arrow) and hard exudates at macula in stellate pattern
Figure 2: Spectral domain optical coherence tomography of right macula showing full thickness macular hole with subretinal fluid with epiretinal membrane (red arrow)
Figure 3: Fundus of the right eye 6 weeks after treatment with oral prednisolone showing disc pallor (blue arrow) and a larger macular hole with subretinal fluid (red arrow)
Oman Journal of Ophthalmology, Vol. 8, No. 1, 2015
Figure 4: Spectral domain optical coherence tomography of the right macula at 6 weeks showing an increase in size of macular hole with persistent epiretinal membrane (red arrow)
45
Seth, et al.: Macular hole in neuroretinitis in an 11-year-old girl rheumatoid arthritis after cataract extraction.[6,10] Three cases of full thickness macular hole have also been described in B. Henselae neuroretinitis.[6-8] The pathogenesis of macular hole in inflammatory diseases is not well-understood. It has been hypothesized that the contraction of premacular vitreous due to inflammation causes tangential traction causing a full-thickness macular hole.[6,10] A lamellar macular hole has also been seen to develop after cystoid macular edema in inflammatory diseases. Three cases have been described where patients presented with neuroretinitis and developed macular hole over a follow up of 1-2 months. To the best of our knowledge, no case has been reported with neuroretinitis and full thickness macular hole at presentation, with a history as short as 2 weeks. The macular hole in our patient was also seen to increase in size over the follow up. This probably represents a progressive vitreous traction and the inflammatory membrane contraction despite control of disease and visual improvement. Hence, all cases with neuroretinitis should be evaluated and followed up for development of full-thickness macular hole.
References 1.
46
Carithers HA. Cat-scratch disease. An overview based on a study of 1,200 patients. Am J Dis Child 1985;139:1124-33.
2. 3.
4.
5.
6. 7. 8.
9.
10.
Ormerod LD, Dailey JP. Ocular manifestations of cat-scratch disease. Curr Opin Ophthalmol 1999;10:209-16. Ormerod LD, Skolnick KA, Menosky MM, Pavan PR, Pon DM. Retinal and choroidal manifestations of cat-scratch disease. Ophthalmology 1998;105:1024-31. Solley WA, Martin DF, Newman NJ, King R, Callanan DG, Zacchei T, et al. Cat scratch disease: Posterior segment manifestations. Ophthalmology 1999;106:1546-53. Cunningham ET Jr, McDonald HR, Schatz H, Johnson RN, Ai E, Grand MG. Inflammatory mass of the optic nerve head associated with systemic Bartonella henselae infection. Arch Ophthalmol 1997;115:1596-7. Albini TA, Lakhanpal RR, Foroozan R, Holz ER. Macular hole in cat scratch disease. Am J Ophthalmol 2005;140:149-51. Donnio A, Jean-Charles A, Merle H. Macular hole following Bartonella henselae neuroretinitis. Eur J Ophthalmol 2008;18:456-8. Alterman MA, Young BK, Eggenberger ER, Kaufman DI. Macular hole: A rare complication of ocular bartonellosis. J Neuroophthalmol 2013;33:153-4. Metz CH, Buer J, Bornfeld N, Lipski A. Bilateral Bartonella henselae neuroretinitis with stellate maculopathy in a 6-year-old boy. Infection 2012;40:191-4. Nussenblatt RB. Macular alterations secondary to intraocular inflammatory disease. Ophthalmology 1986;93:984-8.
Cite this article as: Seth A, Raina UK, Thirumalai S, Batta S, Ghosh B. Fullthickness macular hole in Bartonella henselae neuroretinitis in an 11-year-old girl. Oman J Ophthalmol 2015;8:44-6. Source of Support: Nil, Conflict of Interest: None declared.
Oman Journal of Ophthalmology, Vol. 8, No. 1, 2015
Copyright of Oman Journal of Ophthalmology is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
