Multimodal imaging of torpedo maculopathy in a Chinese woman: a case report

preprint OA: closed
Full text JSON View at publisher

Abstract

Background: Torpedo maculopathy is a rare, benign, and congenital macular lesion that typically appears in a ‘torpedo-shape’ and is located at the temporal macula region. This study aimed to describe in detail regarding torpedo maculopathy in a Chinese woman using multimodal imaging. Case presentation: A 30-year-old Chinese woman with occasional yellowish-white macular lesions in her right eye during a routine examination was presented to our hospital. She had no other symptoms, and the best-corrected visual acuity of both eyes was 6/6. Funduscopic examination revealed a torpedo-shaped and mild hypopigmented lesion in the temporal macular area of her right eye. Infrared fundal (IR) images showed visible lesion contour, transverse elliptical, and with a tip pointing towards the central fovea of the macula. Microperimetry visual field appeared normal. The spectral-domain optical coherence tomography (SD-OCT) showed a normal inner retina, with mild thinner outer retina and retinal pigment epithelium in the temporal macular area, and correspondingly increased choroidal reflectivity. Other OCT findings included outer retinal loss/attenuation with significant atrophy of an intact ellipsoid zone. OCT angiography (OCTA) of choroid capillary layer revealed increased density of choroidal vasculature in corresponding to the area of the lesion, while the superficial and deep layers revealed normal vasculature. Fundus autofluorescence (FAF) revealed normal signal with slight hyperautofluorescence at the nasal lesion margin. Fundus fluorescence angiography (FFA) of the lesion showed variegated fluorescence and no leakage and change in the morphology during the whole imaging process. Conclusions: This is the first report to include a thorough and detailed description of torpedo maculopathy by using fundal photograph, IR, microperimetry visual field, OCT, OCTA, FAF, and FFA. Multimodal imaging provides precious and detailed information to further clarify the characteristics and development of this rare disease.
Full text 55,033 characters · extracted from preprint-html · click to expand
Multimodal imaging of torpedo maculopathy in a Chinese woman: a case report | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case report Multimodal imaging of torpedo maculopathy in a Chinese woman: a case report Yuhua Ding, Bangtao Yao, Hui Ye, Yan Yu This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.2.373/v3 This work is licensed under a CC BY 4.0 License Status: Published Journal Publication published 19 Jul, 2019 Read the published version in BMC Ophthalmology → Version 3 posted 4 You are reading this latest preprint version Show more versions Abstract Background: Torpedo maculopathy is a rare, benign, and congenital macular lesion that typically appears in a ‘torpedo-shape’ and is located at the temporal macula region. This study aimed to describe in detail regarding torpedo maculopathy in a Chinese woman using multimodal imaging. Case presentation: A 30-year-old Chinese woman with occasional yellowish-white macular lesions in her right eye during a routine examination was presented to our hospital. She had no other symptoms, and the best-corrected visual acuity of both eyes was 6/6. Funduscopic examination revealed a torpedo-shaped and mild hypopigmented lesion in the temporal macular area of her right eye. Infrared fundal (IR) images showed visible lesion contour, transverse elliptical, and with a tip pointing towards the central fovea of the macula. Microperimetry visual field appeared normal. The spectral-domain optical coherence tomography (SD-OCT) showed a normal inner retina, with mild thinner outer retina and retinal pigment epithelium in the temporal macular area, and correspondingly increased choroidal reflectivity. Other OCT findings included outer retinal loss/attenuation with significant atrophy of an intact ellipsoid zone. OCT angiography (OCTA) of choroid capillary layer revealed increased density of choroidal vasculature in corresponding to the area of the lesion, while the superficial and deep layers revealed normal vasculature. Fundus autofluorescence (FAF) revealed normal signal with slight hyperautofluorescence at the nasal lesion margin. Fundus fluorescence angiography (FFA) of the lesion showed variegated fluorescence and no leakage and change in the morphology during the whole imaging process. Conclusions: This is the first report to include a thorough and detailed description of torpedo maculopathy by using fundal photograph, IR, microperimetry visual field, OCT, OCTA, FAF, and FFA. Multimodal imaging provides precious and detailed information to further clarify the characteristics and development of this rare disease. Internal Medicine Specialties Torpedo maculopathy multimodal imaging RPE OCTA Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Background Torpedo maculopathy is a rare, benign, and congenital retinal pigment epithelial (RPE) disease, which typically appears as a ‘torpedo-shaped’ lesion in the temporal macula. In 1992, it was first reported by Roseman and Gass [1] as an asymptomatic, solitary hypopigmented nevus of the retinal pigment epithelium. In the classic fundus, it manifests as a solitary hypo-pigmented lesion that is oval in shape, resembling a ‘bullet’ or ‘torpedo,’ with a wedge-shaped tail extending outward and pointing towards the foveola along the horizontal raphe [2]. In 1993, torpedo maculopathy was named by Daily [3] due to its typical appearance. The typical fundus can be distinguished from other lesions, such as toxoplasma scar, traumatic injury, congenital hypertrophy of the RPE (CHRPE), and congenital RPE hypertrophia associated with Gardner syndrome, allowing for diagnosis. However, the etiology of torpedo maculopathy remained unknown. Many imaging devices have been used to observe the lesion. We herein used multimodal imaging for describing torpedo maculopathy in a Chinese woman. To our knowledge, this is the first report in the world to observe torpedo maculopathy simultaneously by using fundal photographs, IR, microperimetry visual field, OCT, OCTA, FAF, and FFA. We hypothesized that this case might be in a very early stage or a mild type of torpedo maculopathy. Case Presentation A 30-year-old Chinese woman with yellowish-white macular lesion in ight eye during a routine examination presented to our hospital. She had no other symptoms. There was no pain or vision loss in her right eye. The patient denied of any traumatic history. Her past medical history and ophthalmic history were negative. The initial best-corrected visual acuity (BCVA) was 6/6 for both eyes. The cornea was clear, and the anterior segment was normal. Pupils were equal, round and reactive to light with no afferent pupillary defect. There was no cataract in both eyes. The initial intraocular pressure (IOP) was 14mmHg in the right eye and 13 mmHg in the left eye. Funduscopic examination of the left eye was unremarkable. A spindle-shaped yellowish-white and hypo-pigmented lesion of about 0.5 disc diameter vertically and by 1 disc diameter horizontally was located in the temporal macular area with a tip pointing towards the central fovea of the macula (Figure 1, A). IR photograph showed that the contour of the lesion was visible, and transverse elliptical and was consistent with the colorful fundus photographs (Figure 1, B). Microperimetry visual field was basically normal (Figure 2). The SD-OCT showed a normal inner retina, mildly thinned outer retina and RPE in the temporal macular area, with correspondingly increased choroidal reflectivity (Figure 3). Other OCT findings included outer retinal loss/attenuation with significant atrophy of an intact ellipsoid zone. OCT angiography (OCTA) of choroid capillary layer revealed increased density of the choroidal vasculature, corresponding to the area of the lesion, while the superficial and deep layers revealed normal (Figure4). With FAF, the lesion showed most of the normal signals with slight hyperautofluorescence at the nasal lesion margin (Figure 5, A). FFA of the lesion showed variegated fluorescence and no leakage and change in the morphology during the whole imaging process (Figure 5, B,C,D). Based on these findings, the patient was confirmed to have torpedo maculopathy clearly. The treatment for this patient was closely observed. The patient was followed-up every 3 to 6 months. Her BCVA remained still 6/6 in both eyes, and the funds lesion remained intact during 15 months followed-up. Discussion and Conclusions Torpedo maculopathy is a rare, benign, and congenital maculopathy, which is usually asymptomatic, but occasionally found during routine examination. It often occurs in patients without any relevant medical history and is most commonly unilateral, although bilateral cases have been reported previously[4]. The torpedo maculopathy appears as a transverse oval, yellowish-white hypo-pigmented lesion, and is located in the temporal macular area, with a tip pointing towards the central fovea. Till now, the etiology of this disease is still unclear. Few studies have been reported to explain the pathogenesis of the lesion. Pian et al[5] assumed that it might be a developmental defect within the nerve-fiber layer at the horizontal raphe. Shields[6] suggested a persistent defect in the development of RPE during the fetal temporal bulge, which might be the reason for the cause of the lesion. Golchet et al[7] hypothesized that the lesion may be related to dysmorphia of the emissary canal of the long posterior ciliary artery and nerve. Microperimetry precisely revealed the correlation of retinal sensitivity and fundus lesion, and also detected microscotoma within the central visual field that may not be detectable by using standard perimetry methods[8]. Published reports have demonstrated that scotoma is frequently associated with torpedo lesion[9-11]. Focal RPE atrophy results showed that reduced metabolites and oxygen supply for the inner retina, secondary choriocapillaris loss and photoreceptor degeneration are associated with the reduction of retinal sensitivity[12]. This case was different from the earlier reports, and there was no microscotoma seen, the retinal sensitivity appeared normal, and the patient had good visual function. Therefore, we speculated that the degree of RPE atrophy was mild in this case, and does not harm the function of overlying photoreceptors. Using SD-OCT, the outer retina was disorganized, and showed significant atrophy of RPE with an intact ellipsoid zone in this case. We thought that the RPE still preserved the function of ingesting photoreceptor cell outer segment, so the ellipsoid zone still remained intact and the patient has preserved the normal visual function. Evan et al[13] identified two patterns of abnormalities: type 1, attenuation of outer retinal structures without outer retinal cavitation; and type 2, those with both attenuation of outer retinal structures and outer retinal cavitation. According to their theory, this patient was included under type 1 torpedo maculopathy. Besides, they also observed that the patients with type 1 lesion (age, 4–37) tended to be younger than those with type 2 lesion (age, 13–73) [13]. This patient was aged 30, and was consistent with the characteristics summarized by them. OCTA non-invasively detects the movement of red blood cells to reveal the retinal and choroidal vascular system. Therefore, alterations in the choriocapillaris can be visualized by using OCTA. Papastefanou and his colleagues[14] described the OCTA features of torpedo lesions with OCTA, and revealed choroidal vascular segmentation with hypo-reflectivity (atrophy), which in turn was correlated to the OCT of the subretinal cleft. While there was no subretinal cleft in our patient, and the OCTA findings were different. OCTA choroid capillary segment revealed increased density of choroidal vasculature, revealing thinner RPE as the optical signal transmission for the increased choroidal thickness; however, the superficial and deep layers were normal. Comparison of our case with the previous reported cases[14] revealed that our case had an early stage according to the OCT classification. Autofluorescence signal is predominantly derived from lipofuscin within the RPE[15]. In our case, FAF showed normal signals mostly with slight hyperautofluorescence at the nasal lesion. The possible explanation for this was due to attenuation of both RPE and outer nuclear layer. FFA of the lesion showed variegated fluorescence and no leakage and change in the morphology during the whole imaging process. This demonstrated no choroidal neovascularization (CNV), while few cases showed the existence of CNV[16]. Lesions in our case were still limited to RPE without CNV. Differential diagnosis such as posterior uveitis should be carefully addressed. Posterior uveitis is also known as choroiditis, and is characterized by vitreous exudation and choroidal vasodilatation, resulting in CNV and visual reduction. Examinations showed by OCTA, FFA and FAF in posterior uveitis are distinguished from torpedo maculopathy. Early hypo-fluorescence followed by late leakage on FFA was observed in active choroiditis lesions. However, healed lesions represented hypo-fluorescence during the early phase with staining but no leakage in the late phase. According to FAF, ill-defined hyper-autofluorescence was observed in active choroiditis lesions, while rounded edges and hypo-autofluorescence within the lesion were shown in healed choroiditis[17]. These are quite different from the torpedo maculopathy. However, there are several limitations in this study. The sample size of the study was small, and indocyanine green angiography was not performed, which helps in better understanding of the differential diagnosis of this disease. Besides, no further follow-up examination is a conceivable limitation of this study. In conclusion, a case of torpedo maculopathy by using fundal photographs, IR, microperimetry visual field, OCT, OCTA, FAF, and FFA simultaneously has been presented for the first time. It is in a very early stage or a mild type of torpedo maculopathy. The natural development of torpedo maculopathy is still unclear, whether it does not develop or develops very slowly. Multimodal imaging provides precious and detailed information for easily diagnosing and deeply understanding this rare disease. This disease is very rare, and needs more case reports worldwide and longer follow-up time to further understand the etiology, characteristics and development of this lesion. Abbreviations BCVA: best-corrected visual acuity; CNV: choroidal neovascularization; CHRPE: congenital hypertrophy of the RPE; FAF: fundus autofluorescence; FFA: Fundus fluorescence angiography; IOP: initial intraocular pressure; IR: Infrared fundal; OCTA: OCT angiographic; RPE: retinal pigment epithelium; SD-OCT: spectral-domain optical coherence tomographic Declarations Funding Not applicable. Availability of data and materials All data and materials of the case are included in this published article. Authors’ contributions Dr. Ding collected the data of the patient, consulted literatures and wrote the manuscript; Dr. Yao edited the manuscript, and dealt with the figures. Dr. Ye also gave useful suggestions on writing and improved the manuscript. Dr. Yu was the Consultant in charge for the case, established the diagnosis and approved the submitted version. All authors read and approved the final manuscript. Acknowledgement Not applicable. Ethics approval and consent to participate Not applicable. Consent for publication Informed written consent had been obtained from the patient. Competing interests The authors declare that they have no competing interests. References 1. Roseman RL, Gass JD. Solitary hypopigmented nevus of the retinal pigment epithelium in the macula. Arch Ophthalmol.1992;110(10):1358-9. 2. Trevino R, Kiani S, Raveendranathan P. The expanding clinical spectrum of torpedo maculopathy. Optom Vis Sci. 2014; 91(4): S71–8. 3. Golchet PR, Jampol LM, Mathura JR, et al. Torpedo maculopathy. Br J Ophthalmol. 2010;94(3):302-6. 4. Richez F, Gueudry J, Brasseur G et al. Bilateral torpedo maculopathy. J Fr Ophtalmol. 2010;33(4):296. 5. Pian D, Ferrucci S, Anderson SF, Wu C. Paramacular coloboma. Optom Vis Sci. 2003;80(8):556-63. 6. Shields CL, Guzman JM, Shapiro MJ, Fogel LE et al. Torpedo maculopathy at the site of the fetal ‘‘bulge’’. Arch Ophthalmol. 2010;128(4):499-501 7. Golchet PR, Jampol LM, Mathura JR, Jr et al. Torpedo maculopathy. Br J Ophthalmol. 2010 Mar;94(3):302-6 8. Sunness JS, Schuchard RA, Shen N et al. Landmark-driven fundus perimetry using the scanning laser ophthalmoscope. Invest Ophthalmol Vis Sci. 1995 Aug;36(9):1863-74 9. Su Y, Gurwood AS. Neurosensory retinal detachment secondary to torpedo maculopathy. Optometry. 2010;81(8):405-7. 10. Sanabria MR, Coco RM, Sanchidrian M. OCT findings in torpedo maculopathy. Retin Cases Brief Rep. 2008;2(2):109-11. 11. Barbazetto IA, Maris PJ, Jr., Greenstein VC. Solitary albinotic spot of the retinal pigment epithelium: a functional and imaging study. Klin Monbl Augenheilkd 2008;225(4):295-7. 12. Midena E, Vujosevic S, Convento E, et al. Microperimetry and fundus autofluo- rescence in patients with early age-related macular degeneration. Br J Ophthalmol . 2007;91(11):1499-503. 13. Wong EN, Fraser-Bell S, Hunyor AP, et al. Novel optical coherence tomography classification of torpedo maculopathy. Clin Experiment Ophthalmol, 2015; 43(4): 342–8. 14. Papastefanou VP, Vázquez-Alfageme C, Keane PA et al. Multimodal imaging of torpedo maculopathy with swept-source, en face optical coherence tomography and optical coherence tomography angiography. Retin Cases Brief Rep. 2018;12(2):153-7. 15. von Rückmann A, Fitzke FW, Bird AC. Distribution of fundus autofluorescence with a scanning laser ophthalmoscope. Br J Ophthalmol .1995;79(5):407-12 16. D. Jurjevic, C. Böni, D. Barthelmes D, et al. Torpedo Maculopathy Associated with Choroidal Neovascularization. Klin Monatsbl Augenheilkd. 2017; 234(4): 508–14. 17. Aggarwal K, Agarwal A, Sharma A, et al. Detection of type 1 choroidal neovascular membranes using optical coherence tomography angiography in tubercular posterior uveitis. Retina. 2018 Apr 23 Cite Share Download PDF Status: Published Journal Publication published 19 Jul, 2019 Read the published version in BMC Ophthalmology → Version 3 posted Editorial decision: Accept 05 Jul, 2019 Editor assigned by journal 03 Jul, 2019 Submission checks completed at journal 02 Jul, 2019 Editor invited by journal 02 Jul, 2019 You are reading this latest preprint version Show more versions Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-373","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case report","associatedPublications":[],"authors":[{"id":107199,"identity":"f507b916-796b-4cc9-87c0-40a1ba2e7b03","order_by":1,"name":"Yuhua Ding","email":"","orcid":"","institution":"JIANGSU PROVINCE HOSPITAL;THE FIRST AFFILIATE HOSPITAL OF NANJING MEDICAL UNIVERSITY","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Yuhua","middleName":"","lastName":"Ding","suffix":""},{"id":107200,"identity":"b561b0f8-4b00-49d2-8d50-bccb272c3816","order_by":2,"name":"Bangtao Yao","email":"","orcid":"","institution":"LISHUI DISTRICT PEOPLE'S HOSPITAL","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Bangtao","middleName":"","lastName":"Yao","suffix":""},{"id":107201,"identity":"57548d8b-71fe-4f68-a0f6-a38bc1c619e2","order_by":3,"name":"Hui Ye","email":"","orcid":"","institution":"JIANGSU PROVINCE HOSPITAL;THE FIRST AFFILIATE HOSPITAL OF NANJING MEDICAL UNIVERSITY","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Hui","middleName":"","lastName":"Ye","suffix":""},{"id":107202,"identity":"e6612094-d1f5-4efa-9912-8cbc477e29e2","order_by":4,"name":"Yan Yu","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA0UlEQVRIiWNgGAWjYPACNgYGCeYDBz78IE0LW+LBmT0kWSTBY3yYg40IhQY3cgw/FzDw2ZtL93w4zMDDIM8vdoCgFmPpGQxsiTvnnN1wuMCCwXDm7ASCWgykeRjYEgxu5G44PIOHIcHgNmEtxr+BWuyBjAeHediI02IGsoVxw40cBuK0SJ55VmbNA/LLjDQDYCBLEPYL3/Hkzbd5GI7Zm0skP/7w4YeNPL80AS0KBzgMGBj/HWMwgPAl8CsHAfkG9gdAqgamZRSMglEwCkYBJgAAbphDyjpwAmsAAAAASUVORK5CYII=","orcid":"","institution":"THE FIRST AFFILIATE HOSPITAL OF NANJING MEDICAL UNIVERSITY","correspondingAuthor":true,"submittingAuthor":false,"prefix":"","firstName":"Yan","middleName":"","lastName":"Yu","suffix":""}],"badges":[],"createdAt":"2019-02-19 14:41:52","currentVersionCode":3,"declarations":"","doi":"10.21203/rs.2.373/v3","doiUrl":"https://doi.org/10.21203/rs.2.373/v3","draftVersion":[],"editorialEvents":[{"content":"https://doi.org/10.1186/s12886-019-1161-7","type":"published","date":"2019-07-19T12:00:00+00:00"}],"editorialNote":"","failedWorkflow":false,"files":[{"id":2612270,"identity":"17ee57fc-db82-4d3a-97bc-356ae2e54993","added_by":"b0e95e7b-bbe0-4bfd-bf12-a325b7db0c3e","created_at":"2020-09-25 20:52:50","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":1428322,"visible":true,"origin":"","legend":"In the right eye, a spindle-shaped yellowish-white and hypo-pigmented lesion, which was about 0.5 disc diameter vertically by 1 disc diameter horizontally, was located in the temporal macular area with a tip pointed towards the central fovea of the macula (Figure 1, A). IR photograph showed that the contour of the lesion was visible, and transverse elliptical and was consistent with the colorful fundus photograph (Figure 1, B).","description":"","filename":"figure1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-373/v3/figure_1.jpg"},{"id":2612269,"identity":"05c445e7-2337-42ab-a2e0-51457ae6c751","added_by":"dbe1c910-fa3f-4a3a-a5c6-af99624d3e99","created_at":"2020-09-25 20:52:50","extension":"jpg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":1107167,"visible":true,"origin":"","legend":"Microperimetry visual field was basically normal.","description":"","filename":"figure2.jpg","url":"https://assets-eu.researchsquare.com/files/rs-373/v3/figure_2.jpg"},{"id":2612271,"identity":"499f6e24-2591-4ac7-92e8-f41a456cca7e","added_by":"dbe1c910-fa3f-4a3a-a5c6-af99624d3e99","created_at":"2020-09-25 20:52:50","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":1091742,"visible":true,"origin":"","legend":"The SD-OCT showed a normal inner retina, mildly thinned outer retina and RPE in the temporal macular area, with correspondingly increased choroidal reflectivity.","description":"","filename":"figure3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-373/v3/figure_3.jpg"},{"id":2612272,"identity":"74b96d74-ca29-41bc-a673-f7660a47b404","added_by":"b0e95e7b-bbe0-4bfd-bf12-a325b7db0c3e","created_at":"2020-09-25 20:52:50","extension":"jpg","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":2751628,"visible":true,"origin":"","legend":"OCT angiography (OCTA) of choroid capillary segment revealed increased density of the choroidal vasculature, corresponding to the area of the lesion (Figure4, A). OCTA of deep retinal layer(Figure4, B) and superficial retinal layer(Figure4, C) revealed normal.","description":"","filename":"figure4.jpg","url":"https://assets-eu.researchsquare.com/files/rs-373/v3/figure_4.jpg"},{"id":2612273,"identity":"d61bfaaf-5abb-4f0c-8be1-cc5c3ff8f68d","added_by":"b0e95e7b-bbe0-4bfd-bf12-a325b7db0c3e","created_at":"2020-09-25 20:52:50","extension":"jpg","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":1871649,"visible":true,"origin":"","legend":"With FAF, the lesion showed slight hypoautofluorescene with marginal mild hyperautofluorescence (Figure 5, A). FFA of the lesion showed variegated fluorescence and no leakage and change in the morphology during the whole imaging process (Figure 5, B,C,D).","description":"","filename":"figure5.jpg","url":"https://assets-eu.researchsquare.com/files/rs-373/v3/figure_5.jpg"},{"id":13469829,"identity":"a72d12ac-e695-4336-97f8-65097de5c346","added_by":"auto","created_at":"2021-09-16 21:03:20","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":913917,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-373/v3/9519a793-2b24-4c5e-bc73-fe0b845870aa.pdf"}],"financialInterests":"","formattedTitle":"Multimodal imaging of torpedo maculopathy in a Chinese woman: a case report","fulltext":[{"header":"Background","content":"\u003cp\u003eTorpedo maculopathy is a rare, benign, and congenital retinal pigment epithelial (RPE)\n disease, which typically appears as a ‘torpedo-shaped’ lesion in the temporal macula.\n In 1992, it was first reported by Roseman and Gass [1] as an asymptomatic, solitary\n hypopigmented nevus of the retinal pigment epithelium. In the classic fundus, it manifests\n as a solitary hypo-pigmented lesion that is oval in shape, resembling a ‘bullet’ or\n ‘torpedo,’ with a wedge-shaped tail extending outward and pointing towards the foveola\n along the horizontal raphe [2]. In 1993, torpedo maculopathy was named by Daily [3]\n due to its typical appearance. The typical fundus can be distinguished from other\n lesions, such as toxoplasma scar, traumatic injury, congenital hypertrophy of the\n RPE (CHRPE), and congenital RPE hypertrophia associated with Gardner syndrome, allowing\n for diagnosis. \u003c/p\u003e\n \n\u003cp\u003eHowever, the etiology of torpedo maculopathy remained unknown. Many imaging devices\n have been used to observe the lesion. We herein used multimodal imaging for describing\n torpedo maculopathy in a Chinese woman. To our knowledge, this is the first report\n in the world to observe torpedo maculopathy simultaneously by using fundal photographs,\n IR, microperimetry visual field, OCT, OCTA, FAF, and FFA. We hypothesized that this\n case might be in a very early stage or a mild type of torpedo maculopathy.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003eA 30-year-old Chinese woman with yellowish-white macular lesion in ight eye during\n a routine examination presented to our hospital. She had no other symptoms. There\n was no pain or vision loss in her right eye. The patient denied of any traumatic history.\n Her past medical history and ophthalmic history were negative. The initial best-corrected\n visual acuity (BCVA) was 6/6 for both eyes. The cornea was clear, and the anterior\n segment was normal. Pupils were equal, round and reactive to light with no afferent\n pupillary defect. There was no cataract in both eyes. The initial intraocular pressure\n (IOP) was 14mmHg in the right eye and 13 mmHg in the left eye. Funduscopic examination\n of the left eye was unremarkable. A spindle-shaped yellowish-white and hypo-pigmented\n lesion of about 0.5 disc diameter vertically and by 1 disc diameter horizontally was\n located in the temporal macular area with a tip pointing towards the central fovea\n of the macula (Figure 1, A). IR photograph showed that the contour of the lesion was\n visible, and transverse elliptical and was consistent with the colorful fundus photographs\n (Figure 1, B). Microperimetry visual field was basically normal (Figure 2). The SD-OCT\n showed a normal inner retina, mildly thinned outer retina and RPE in the temporal\n macular area, with correspondingly increased choroidal reflectivity (Figure 3). Other OCT findings included outer retinal loss/attenuation with significant atrophy\n of an intact ellipsoid zone. OCT angiography (OCTA) of choroid capillary layer revealed increased density of the\n choroidal vasculature, corresponding to the area of the lesion, while the superficial and deep layers revealed normal (Figure4). With FAF, the lesion showed most of the normal signals with slight hyperautofluorescence\n at the nasal lesion margin (Figure 5, A). FFA of the lesion showed variegated fluorescence and no leakage and\n change in the morphology during the whole imaging process (Figure 5, B,C,D). Based\n on these findings, the patient was confirmed to have torpedo maculopathy clearly.\u003c/p\u003e\n \n\u003cp\u003eThe treatment for this patient was closely observed. The patient was followed-up every\n 3 to 6 months. Her BCVA remained still 6/6 in both eyes, and the funds lesion remained\n intact during 15 months followed-up.\u003c/p\u003e"},{"header":"Discussion and Conclusions","content":"\u003cp\u003eTorpedo maculopathy is a rare, benign, and congenital maculopathy, which is usually\n asymptomatic, but occasionally found during routine examination. It often occurs in\n patients without any relevant medical history and is most commonly unilateral, although\n bilateral cases have been reported previously[4]. The torpedo maculopathy appears\n as a transverse oval, yellowish-white hypo-pigmented lesion, and is located in the\n temporal macular area, with a tip pointing towards the central fovea.\u003c/p\u003e\n \n\u003cp\u003eTill now, the etiology of this disease is still unclear. Few studies have been reported\n to explain the pathogenesis of the lesion. Pian et al[5] assumed that it might be\n a developmental defect within the nerve-fiber layer at the horizontal raphe. Shields[6]\n suggested a persistent defect in the development of RPE during the fetal temporal\n bulge, which might be the reason for the cause of the lesion. Golchet et al[7] hypothesized\n that the lesion may be related to dysmorphia of the emissary canal of the long posterior\n ciliary artery and nerve. \u003c/p\u003e\n \n\u003cp\u003eMicroperimetry precisely revealed the correlation of retinal sensitivity and fundus\n lesion, and also detected microscotoma within the central visual field that may not\n be detectable by using standard perimetry methods[8]. Published reports have demonstrated\n that scotoma is frequently associated with torpedo lesion[9-11]. Focal RPE atrophy\n results showed that reduced metabolites and oxygen supply for the inner retina, secondary\n choriocapillaris loss and photoreceptor degeneration are associated with the reduction\n of retinal sensitivity[12]. This case was different from the earlier reports, and\n there was no microscotoma seen, the retinal sensitivity appeared normal, and the patient\n had good visual function. Therefore, we speculated that the degree of RPE atrophy\n was mild in this case, and does not harm the function of overlying photoreceptors.\u003c/p\u003e\n \n\u003cp\u003eUsing SD-OCT, the outer retina was disorganized, and showed significant atrophy of\n RPE with an intact ellipsoid zone in this case. We thought that the RPE still preserved\n the function of ingesting photoreceptor cell outer segment, so the ellipsoid zone\n still remained intact and the patient has preserved the normal visual function. Evan\n et al[13] identified two patterns of abnormalities: type 1, attenuation of outer retinal\n structures without outer retinal cavitation; and type 2, those with both attenuation\n of outer retinal structures and outer retinal cavitation. According to their theory,\n this patient was included under type 1 torpedo maculopathy. Besides, they also observed\n that the patients with type 1 lesion (age, 4–37) tended to be younger than those with\n type 2 lesion (age, 13–73) [13]. This patient was aged 30, and was consistent with\n the characteristics summarized by them. \u003c/p\u003e\n \n\u003cp\u003eOCTA non-invasively detects the movement of red blood cells to reveal the retinal\n and choroidal vascular system. Therefore, alterations in the choriocapillaris can\n be visualized by using OCTA. Papastefanou and his colleagues[14] described the OCTA\n features of torpedo lesions with OCTA, and revealed choroidal vascular segmentation\n with hypo-reflectivity (atrophy), which in turn was correlated to the OCT of the subretinal\n cleft. While there was no subretinal cleft in our patient, and the OCTA findings were\n different. OCTA choroid capillary segment revealed increased density of choroidal\n vasculature, revealing thinner RPE as the optical signal transmission for the increased\n choroidal thickness; however, the superficial and deep layers were normal. Comparison of our case with the previous reported cases[14] revealed that our case\n had an early stage according to the OCT classification.\u003c/p\u003e\n \n\u003cp\u003eAutofluorescence signal is predominantly derived from lipofuscin within the RPE[15].\n In our case, FAF showed normal signals mostly with slight hyperautofluorescence at the nasal lesion.\n The possible explanation for this was due to attenuation of both RPE and outer nuclear\n layer.\u003c/p\u003e\n \n\u003cp\u003eFFA of the lesion showed variegated fluorescence and no leakage and change in the\n morphology during the whole imaging process. This demonstrated no choroidal neovascularization\n (CNV), while few cases showed the existence of CNV[16]. Lesions in our case were still\n limited to RPE without CNV. \u003c/p\u003e\n \n\u003cp\u003eDifferential diagnosis such as posterior uveitis should be carefully addressed. Posterior\n uveitis is also known as choroiditis, and is characterized by vitreous exudation and\n choroidal vasodilatation, resulting in CNV and visual reduction. Examinations showed\n by OCTA, FFA and FAF in posterior uveitis are distinguished from torpedo maculopathy.\n Early hypo-fluorescence followed by late leakage on FFA was observed in active choroiditis\n lesions. However, healed lesions represented hypo-fluorescence during the early phase\n with staining but no leakage in the late phase. According to FAF, ill-defined hyper-autofluorescence\n was observed in active choroiditis lesions, while rounded edges and hypo-autofluorescence within the lesion were shown\n in healed choroiditis[17]. These are quite different from the torpedo maculopathy.\u003c/p\u003e\n \n\u003cp\u003eHowever, there are several limitations in this study. The sample size of the study\n was small, and indocyanine green angiography was not performed, which helps in better\n understanding of the differential diagnosis of this disease. Besides, no further follow-up\n examination is a conceivable limitation of this study.\u003c/p\u003e\n \n\u003cp\u003eIn conclusion, a case of torpedo maculopathy by using fundal photographs, IR, microperimetry\n visual field, OCT, OCTA, FAF, and FFA simultaneously has been presented for the first\n time. It is in a very early stage or a mild type of torpedo maculopathy. The natural\n development of torpedo maculopathy is still unclear, whether it does not develop or\n develops very slowly. Multimodal imaging provides precious and detailed information\n for easily diagnosing and deeply understanding this rare disease. This disease is\n very rare, and needs more case reports worldwide and longer follow-up time to further\n understand the etiology, characteristics and development of this lesion. \u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cp\u003eBCVA: best-corrected visual acuity; CNV: choroidal neovascularization; CHRPE: congenital hypertrophy of the RPE; FAF: fundus autofluorescence; FFA: Fundus fluorescence angiography; IOP: initial intraocular\n pressure; IR: Infrared fundal; OCTA: OCT angiographic; RPE: retinal pigment epithelium;\n SD-OCT: spectral-domain optical coherence tomographic \u003c/p\u003e"},{"header":"Declarations","content":"\u003ch2\u003eFunding\u003c/h2\u003e\n \n\u003cp\u003eNot applicable.\u003c/p\u003e\n \n\u003ch2\u003eAvailability of data and materials \u003c/h2\u003e\n \n\u003cp\u003eAll data and materials of the case are included in this published article.\u003c/p\u003e\n \n\u003ch2\u003eAuthors’ contributions \u003c/h2\u003e\n \n\u003cp\u003eDr. Ding collected the data of the patient, consulted literatures and wrote the manuscript;\n Dr. Yao edited the manuscript, and dealt with the figures. Dr. Ye also gave useful\n suggestions on writing and improved the manuscript. Dr. Yu was the Consultant in charge\n for the case, established the diagnosis and approved the submitted version. All authors\n read and approved the final manuscript. \u003c/p\u003e\n \n\u003ch2\u003eAcknowledgement\u003c/h2\u003e\n \n\u003cp\u003eNot applicable.\u003c/p\u003e\n \n\u003ch2\u003eEthics approval and consent to participate \u003c/h2\u003e\n \n\u003cp\u003eNot applicable.\u003c/p\u003e\n \n\u003ch2\u003eConsent for publication\u003c/h2\u003e\n \n\u003cp\u003eInformed written consent had been obtained from the patient.\u003c/p\u003e\n \n\u003ch2\u003eCompeting interests\u003c/h2\u003e\n \n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e"},{"header":"References","content":"\u003cp\u003e1. Roseman RL, Gass JD. Solitary hypopigmented nevus of the retinal pigment epithelium\n in the macula. Arch Ophthalmol.1992;110(10):1358-9.\u003c/p\u003e\n \n\u003cp\u003e2. Trevino R, Kiani S, Raveendranathan P. The expanding clinical spectrum of torpedo\n maculopathy. Optom Vis Sci. 2014; 91(4): S71–8. \u003c/p\u003e\n \n\u003cp\u003e3. Golchet PR, Jampol LM, Mathura JR, et al. Torpedo maculopathy. Br J Ophthalmol.\n 2010;94(3):302-6.\u003c/p\u003e\n \n\u003cp\u003e4. Richez F, Gueudry J, Brasseur G et al. Bilateral torpedo maculopathy. J Fr Ophtalmol.\n 2010;33(4):296.\u003c/p\u003e\n \n\u003cp\u003e5. Pian D, Ferrucci S, Anderson SF, Wu C. Paramacular coloboma. Optom Vis Sci. 2003;80(8):556-63.\n \u003c/p\u003e\n \n\u003cp\u003e6. Shields CL, Guzman JM, Shapiro MJ, Fogel LE et al. Torpedo maculopathy at the site of the fetal ‘‘bulge’’. Arch Ophthalmol. 2010;128(4):499-501\u003c/p\u003e\n \n\u003cp\u003e7. Golchet PR, Jampol LM, Mathura JR, Jr et al. Torpedo maculopathy. Br J Ophthalmol.\n 2010 Mar;94(3):302-6 \u003c/p\u003e\n \n\u003cp\u003e8. Sunness JS, Schuchard RA, Shen N et al. Landmark-driven fundus perimetry using the scanning laser ophthalmoscope.\n Invest Ophthalmol Vis Sci. 1995 Aug;36(9):1863-74\u003c/p\u003e\n \n\u003cp\u003e9. Su Y, Gurwood AS. Neurosensory retinal detachment secondary to torpedo maculopathy.\n Optometry. 2010;81(8):405-7.\u003c/p\u003e\n \n\u003cp\u003e10. Sanabria MR, Coco RM, Sanchidrian M. OCT findings in torpedo maculopathy. Retin\n Cases Brief Rep. 2008;2(2):109-11.\u003c/p\u003e\n \n\u003cp\u003e11. Barbazetto IA, Maris PJ, Jr., Greenstein VC. Solitary albinotic spot of the retinal\n pigment epithelium: a functional and imaging study. Klin Monbl Augenheilkd 2008;225(4):295-7.\u003c/p\u003e\n \n\u003cp\u003e12. Midena E, Vujosevic S, Convento E, et al. Microperimetry and fundus autofluo-\n rescence in patients with early age-related macular degeneration. Br J Ophthalmol\n . 2007;91(11):1499-503. \u003c/p\u003e\n \n\u003cp\u003e13. Wong EN, Fraser-Bell S, Hunyor AP, et al. Novel optical coherence tomography classification of torpedo maculopathy.\n Clin Experiment Ophthalmol, 2015; 43(4): 342–8.\u003c/p\u003e\n \n\u003cp\u003e14. Papastefanou VP, Vázquez-Alfageme C, Keane PA et al. Multimodal imaging of torpedo\n maculopathy with swept-source, en face optical coherence tomography and optical coherence\n tomography angiography. Retin Cases Brief Rep. 2018;12(2):153-7.\u003c/p\u003e\n \n\u003cp\u003e15. von Rückmann A, Fitzke FW, Bird AC. Distribution of fundus autofluorescence with\n a scanning laser ophthalmoscope. Br J Ophthalmol .1995;79(5):407-12\u003c/p\u003e\n \n\u003cp\u003e16. D. Jurjevic, C. Böni, D. Barthelmes D, et al. Torpedo Maculopathy Associated with\n Choroidal Neovascularization. Klin Monatsbl Augenheilkd. 2017; 234(4): 508–14.\u003c/p\u003e\n \n\u003cp\u003e17. Aggarwal K, Agarwal A, Sharma A, et al. Detection of type 1 choroidal neovascular membranes using optical coherence\n tomography angiography in tubercular posterior uveitis. Retina. 2018 Apr 23\u003c/p\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bmc-ophthalmology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"boph","sideBox":"Learn more about [BMC Ophthalmology](http://bmcophthalmol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/boph","title":"BMC Ophthalmology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Torpedo maculopathy, multimodal imaging, RPE, OCTA","lastPublishedDoi":"10.21203/rs.2.373/v3","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.2.373/v3","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"Background: Torpedo maculopathy is a rare, benign, and congenital macular lesion that typically appears in a ‘torpedo-shape’ and is located at the temporal macula region. This study aimed to describe in detail regarding torpedo maculopathy in a Chinese woman using multimodal imaging.\nCase presentation: A 30-year-old Chinese woman with occasional yellowish-white macular lesions in her right eye during a routine examination was presented to our hospital. She had no other symptoms, and the best-corrected visual acuity of both eyes was 6/6. Funduscopic examination revealed a torpedo-shaped and mild hypopigmented lesion in the temporal macular area of her right eye. Infrared fundal (IR) images showed visible lesion contour, transverse elliptical, and with a tip pointing towards the central fovea of the macula. Microperimetry visual field appeared normal. The spectral-domain optical coherence tomography (SD-OCT) showed a normal inner retina, with mild thinner outer retina and retinal pigment epithelium in the temporal macular area, and correspondingly increased choroidal reflectivity. Other OCT findings included outer retinal loss/attenuation with significant atrophy of an intact ellipsoid zone. OCT angiography (OCTA) of choroid capillary layer revealed increased density of choroidal vasculature in corresponding to the area of the lesion, while the superficial and deep layers revealed normal vasculature. Fundus autofluorescence (FAF) revealed normal signal with slight hyperautofluorescence at the nasal lesion margin. Fundus fluorescence angiography (FFA) of the lesion showed variegated fluorescence and no leakage and change in the morphology during the whole imaging process.\nConclusions: This is the first report to include a thorough and detailed description of torpedo maculopathy by using fundal photograph, IR, microperimetry visual field, OCT, OCTA, FAF, and FFA. Multimodal imaging provides precious and detailed information to further clarify the characteristics and development of this rare disease.","manuscriptTitle":"Multimodal imaging of torpedo maculopathy in a Chinese woman: a case report","msid":"","msnumber":"","nonDraftVersions":[{"code":3,"date":"2019-07-15 18:23:59","doi":"10.21203/rs.2.373/v3","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Accept","date":"2019-07-05T12:00:00+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2019-07-03T12:00:00+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2019-07-02T12:00:00+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2019-07-02T12:00:00+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-ophthalmology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"boph","sideBox":"Learn more about [BMC Ophthalmology](http://bmcophthalmol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/boph","title":"BMC Ophthalmology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}},{"code":2,"date":"2019-05-16 18:33:05","doi":"10.21203/rs.2.373/v2","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Minor revision","date":"2019-06-29T12:00:00+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2019-06-03T12:00:00+00:00","index":1,"fulltext":"Form responses:\n---\n* Does the work include the necessary controls?\nIf not, please specify which controls are required in your comments to the authors.\tNo: **Are the conclusions drawn adequately supported by the data shown?\nIf not, please explain in your comments to the authors.\tYes**\n"},{"type":"reviewerAgreed","content":"","date":"2019-05-23T12:00:00+00:00","index":2,"fulltext":""},{"type":"reviewerAgreed","content":"","date":"2019-05-19T12:00:00+00:00","index":1,"fulltext":""},{"type":"reviewersInvited","content":"","date":"2019-05-18T12:00:00+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2019-05-15T12:00:00+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2019-05-15T12:00:00+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2019-05-15T12:00:00+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-ophthalmology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"boph","sideBox":"Learn more about [BMC Ophthalmology](http://bmcophthalmol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/boph","title":"BMC Ophthalmology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}},{"code":1,"date":"2019-02-21 12:23:09","doi":"10.21203/rs.2.373/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Major revision","date":"2019-04-21T12:00:00+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2019-04-13T12:00:00+00:00","index":2,"fulltext":"Form responses:\n---\n* Are the methods appropriate and well described?: **Yes**\n* Does the work include the necessary controls?: **Yes**\n* Are the conclusions drawn adequately supported by the data shown?: **Yes**\n* Are you able to assess any statistics in the manuscript or would you recommend an additional statistical review?: **Not relevant to this manuscript**\n* Quality of written English: **Needs some language corrections before being published**\n* Declaration of competing interests: **I declare that I have no competing interests**\n\nComments to Author:\n---\nTitle: multimodal imaging of torpedo maculopathy in a Chinese woman: case report\n\nThis report describes the different imaging and functional findings in a case of torpedo maculopathy.\n\nStrengths\n\nTorpedo maculopathy is a rare congenital defect of the posterior pole with poorly understood pathophysiology with little information on retinal function changes over time.\n\nThis single case report was well structured, starting with the case presentation and history of the patient, followed by systemic clinical examination from anterior segment to the posterior segment.\n\nThe study used a wide variety different imaging modalities and functional testing to evaluate this macular lesion.\n\nA notable contribution from this report is the microperimetry which has not been published in conjunction with OCT angiography in the same case. At the same time there has been numerous papers published on OCT angiography findings alone in this malformation.\n\nComparison between the results of this study with other studies related to the same condition can be helpful for ophthalmologists around the word to understand the different clinical presentation and progression of this rare disease allowing for better management and follow up\n\nWeakness\nNumerous previous case reports and case series has described in details this congenital malformation with OCTA and fundus autofluorescence, this single case report adds microperimetry which is an important addition.\n\nIn the description of the OCT of this case the authors mentions (page 4 line 60) \"disorganization of the outer retina\" the outer retina is not disorganised there is outer retinal loss/attenuation with relative preservation of the IS/OS or ellipsoid zone.\n\nThe FAF findings were attributed to RPE atrophy, I think the FAF pattern over the lesion was mostly of normal signal (not hyper and not hypo) and that's due to attenuation of both RPE and outer nuclear layer. There was slight hyper FAF at the nasal lesion margin and that's probably due to some RPE hyperplasia at that location.\n\nIt would be of extreme importance to provide the FAF and OCT at the start and at the end of the follow up period to compare since there are the 2 tests with obvious abnormalities.\n\nThe provided OCTA only shows the angiography enface image for the choriocapillaris layer. I recommend including the deep and superficial retinal OCT angiography enface image with projection and segmentation artifacts removed. Also preferably to include the structural OCT enface images for the different layers.\n"},{"type":"editorInvitedReview","content":"","date":"2019-04-13T12:00:00+00:00","index":1,"fulltext":"Form responses:\n---\n* Are the methods appropriate and well described?: **Yes**\n* Does the work include the necessary controls?: **Yes**\n* Are the conclusions drawn adequately supported by the data shown?: **Yes**\n* Are you able to assess any statistics in the manuscript or would you recommend an additional statistical review?: **Not relevant to this manuscript**\n* Quality of written English: **Needs some language corrections before being published**\n* Declaration of competing interests: **I declare that I have no competing interests**\n\nComments to Author:\n---\nTitle: multimodal imaging of torpedo maculopathy in a Chinese woman: case report\n\nThis report describes the different imaging and functional findings in a case of torpedo maculopathy.\n\nStrengths\n\nTorpedo maculopathy is a rare congenital defect of the posterior pole with poorly understood pathophysiology with little information on retinal function changes over time.\n\nThis single case report was well structured, starting with the case presentation and history of the patient, followed by systemic clinical examination from anterior segment to the posterior segment.\n\nThe study used a wide variety different imaging modalities and functional testing to evaluate this macular lesion.\n\nA notable contribution from this report is the microperimetry which has not been published in conjunction with OCT angiography in the same case. At the same time there has been numerous papers published on OCT angiography findings alone in this malformation.\n\nComparison between the results of this study with other studies related to the same condition can be helpful for ophthalmologists around the word to understand the different clinical presentation and progression of this rare disease allowing for better management and follow up\n\nWeakness\nNumerous previous case reports and case series has described in details this congenital malformation with OCTA and fundus autofluorescence, this single case report adds microperimetry which is an important addition.\n\nIn the description of the OCT of this case the authors mentions (page 4 line 60) \"disorganization of the outer retina\" the outer retina is not disorganised there is outer retinal loss/attenuation with relative preservation of the IS/OS or ellipsoid zone.\n\nThe FAF findings were attributed to RPE atrophy, I think the FAF pattern over the lesion was mostly of normal signal (not hyper and not hypo) and that's due to attenuation of both RPE and outer nuclear layer. There was slight hyper FAF at the nasal lesion margin and that's probably due to some RPE hyperplasia at that location.\n\nIt would be of extreme importance to provide the FAF and OCT at the start and at the end of the follow up period to compare since there are the 2 tests with obvious abnormalities.\n\nThe provided OCTA only shows the angiography enface image for the choriocapillaris layer. I recommend including the deep and superficial retinal OCT angiography enface image with projection and segmentation artifacts removed. Also preferably to include the structural OCT enface images for the different layers.\n"},{"type":"reviewerAgreed","content":"","date":"2019-04-03T12:00:00+00:00","index":2,"fulltext":""},{"type":"reviewerAgreed","content":"","date":"2019-03-31T12:00:00+00:00","index":1,"fulltext":""},{"type":"reviewersInvited","content":"","date":"2019-03-04T12:00:00+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2019-02-22T12:00:00+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2019-02-19T12:00:00+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2019-02-19T12:00:00+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-ophthalmology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"boph","sideBox":"Learn more about [BMC Ophthalmology](http://bmcophthalmol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/boph","title":"BMC Ophthalmology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"b909687b-e819-452b-b112-85e4e4952f70","owner":[],"postedDate":"July 15th, 2019","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"published-in-journal","subjectAreas":[{"id":12303,"name":"Internal Medicine Specialties"}],"tags":[],"updatedAt":"","versionOfRecord":{"articleIdentity":"rs-373","link":"https://doi.org/10.1186/s12886-019-1161-7","journal":{"identity":"bmc-ophthalmology","isVorOnly":false,"title":"BMC Ophthalmology"},"publishedOn":"2019-07-19 12:00:00","publishedOnDateReadable":"July 19th, 2019"},"versionCreatedAt":"2019-07-15 18:23:59","video":"","vorDoi":"10.1186/s12886-019-1161-7","vorDoiUrl":"https://doi.org/10.1186/s12886-019-1161-7","workflowStages":[]},"version":"v3","identity":"rs-373","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"identity":"rs-373","version":["v3"]},"buildId":"WrCJVZZCHTDjtuVLN7oU0","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}

Text is read by the "Ask this paper" AI Q&A widget below. Extraction quality varies by source — PMC NXML preserves structure cleanly, OA-HTML may include some navigation residue, and OA-PDF can have broken hyphenation. The publisher copy (via DOI) is the canonical version.

My notes (saved in your browser only)

Ask this paper AI returns verbatim quotes from the full text · source: preprint-html

Answers must be backed by verbatim quotes from this paper's full text. Hallucinated quotes are dropped automatically; if no verbatim passage answers the question, we say so. How this works

Citation neighborhood (no data yet)

We don't have any in-corpus citations linked to this paper yet. The paper's references may be in our DB but unresolved to ``paper_id`` (resolution happens at ingest when the cited DOI matches a row we already have). Run the cross-source citation reconcile pass to retry.

Source provenance

europepmc
last seen: 2026-05-19T01:45:01.086888+00:00