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Anterior stromal puncture for the treatment of Brown–McLean syndrome
a r c h s o c e s p o f t a l m o l . 2 0 1 3;8 8(5):193–196
ARCHIVOS DE LA SOCIEDAD ESPAÑOLA DE OFTALMOLOGÍA www.elsevier.es/oftalmologia
Short communication
Anterior stromal puncture for the treatment of Brown–McLean syndrome夽,夽夽 P. Rodríguez-Ausín ∗ , K. Pachkoria Clínica Oftalmológica Suárez Leoz, Madrid, Spain
a r t i c l e
i n f o
a b s t r a c t
Article history:
Case report: The Brown–McLean syndrome is defined by peripheral corneal edema sparing
Received 18 December 2011
the central cornea. We report a patient with bullous annular keratopathy following pha-
Accepted 8 April 2012
coemulsification and implantation of posterior chamber intraocular lens. The uncorrected
Available online 28 June 2013
and best corrected visual acuity was 20/30. The patient required the use of a bandage contact lens for almost 5 years to relieve the ocular discomfort.
Keywords:
Discussion: Anterior stromal puncture was performed using a cystotome. Two weeks after
Peripheral corneal edema
the procedure the ocular discomfort disappeared and the use of bandage contact lens was
Anterior stromal puncture
no longer required. Anterior stromal puncture is a simple procedure that is performed under
Bullous keratopathy
the slit lamp, and can alleviate the symptoms in patients with Brown–McLean syndrome.
Brown–McLean syndrome
© 2011 Sociedad Española de Oftalmología. Published by Elsevier España, S.L. All rights reserved.
Keratoplasty
Micropunción estromal anterior como tratamiento del síndrome de Brown–McLean r e s u m e n Palabras clave:
El síndrome Brown–McLean consiste en la presencia de edema corneal periférico respetando
Edema corneal periférico
˜ la córnea central. Presentamos un paciente con queratopatia bullosa anular anos después
Punción estromal anterior
de facoemulsificación e implante de lente en cámara posterior. Su agudeza visual espon-
Queratopatía bullosa
tánea y corregida era 20/30 y precisaba lente de contacto blanda para aliviar molestias desde
Síndrome de Brown–McLean
˜ hacía 5 anos. Practicamos micropunción estromal anterior con cistitomo, y en 2 semanas
Queratoplastia
las molestias desaparecieron y no precisa porte de lente blanda. La micropunción estromal es un procedimiento simple que se realiza en lámpara de hendidura y que puede eliminar la sintomatología del síndrome de Brown–McLean. © 2011 Sociedad Española de Oftalmología. Publicado por Elsevier España, S.L. Todos los derechos reservados.
夽 Please cite this article as: Rodríguez-Asuín P, Pachkoria K. Micropunción estromal anterior como tratamiento del síndrome de Brown–McLean. Arch Soc Esp Oftalmol. 2013;88:193–6. 夽夽 ˜ Presented as a verbal communication at the 87.◦ Congreso de la Sociedad Espanola de Oftalmología, Oviedo, 2011. ∗ Corresponding author. E-mail address: [email protected] (P. Rodríguez-Ausín).
2173-5794/$ – see front matter © 2011 Sociedad Española de Oftalmología. Published by Elsevier España, S.L. All rights reserved.
194
a r c h s o c e s p o f t a l m o l . 2 0 1 3;8 8(5):193–196
Introduction The Brown–McLean syndrome (BMS) consists in the appearance of peripheral corneal edema sparing the central area. It was first described in 19691 and is generally associated to previous intracapsular lens surgery. BMS patients are usually asymptomatic although some documented cases exhibited chronic irritation symptoms, pain and septic corneal ulcers. Various treatment options with varying degrees of success have been published for symptomatic patients including hypertonic saline solutions or lubricants,2 extended use soft contact lenses, penetrating keratoplasty, removal of the phakic lens in the anterior chamber and anterior stromal micropuncture.3 This paper presents a symptomatic BMS case successfully treated with anterior stromal micropuncture (ASM).
Case report Male patient, 72, intervened for phacoemulsification with intraocular lens implant in both eyes 12 years before, also requiring vitrectomy for removing nuclear remains in the right eye. The patient had to use soft contact lenses starting 5 years before with substitution every 2 months due to persistent discomfort in the right eye (RE). Non-corrected and best corrected visual acuity was of 20/30 in both eyes (BE). Biomicroscopy revealed pseudophakia in the anterior chamber of BE and a 3 mm peripheral annular corneal edema in the RE sparing the central cornea with peripheral endothelial pigment dispersion (Fig. 1A and B). No gutattas were observed in BE. The LE cornea did not exhibit pathological findings. Intraocular pressure (IOP) was normal in BE and central endothelial cell density measured with SP-3000P TOPCON® mirror microscope was of 1300 and 1500 cells/mm2 in RE and LE respectively. The peripheral density was not displayed due to edema in RE. In April 2011, ASM was performed in slit lamp under topical anesthesia with 0.5% tetracaine (Colircusí Anestésico® , Alcon-Cusí, Barcelona, Spain), consisting in 40–50 superficial micropunctures (one fourth thickness, approximately 150 m) in each quadrant utilizing a 25 G cystotome (BD VisitecTM , Waltham, USA) sparing the central cornea (Fig. 2A and B). Antibiotic prophylaxis was applied with moxifloxacin (Vigamox® , Alcon-Cusí, El Masnou, Barcelona, Spain) before and after the procedure. 2 weeks later, the patient was able to stop using the contact lens undisturbed and in 3 weeks subepithelial fibrosis was observed in the treated area. The patient remains asymptomatic after 12 months of follow-up (Fig. 3A and B).
Discussion BMS consists in peripheral corneal edema with circumferential progression which typically spares the central cornea. In addition, dotted brownish-orange pigmentation may appear in the peripheral endothelium. Central endothelial density is somewhat below normal values and cornea gutatta may coexist. It has been described mainly after intracapsular lens extraction and with less frequency after extracapsular
Fig. 1 – (A) Cornea with blurred ring and presence of soft contact lens and (B) peripheral edema with pigmented deposits.
extraction, phacoemulsification, keratoplasty, IOL implant in CA and vitrectomy. In addition, it has been reported in the absence of surgery associated to ocular traumatism, closed angle glaucoma, myotonic dystrophy and lens subluxation.3,4 Suggested pathogenics include iris peripheral endothelial traumatism, IOL haptics, dislocated vitreous or lens which causes endothelial abrasion and cell loss in the periphery, without discarding associated genetic predisposition.4 The period of time in which BMS appears after surgery has been reported between 1.5 and 34 years. In the instant case it appeared after 10 years and unilaterally. This extended period of time has made us consider progressive RE endothelial damage without knowing the intra- and post surgery details of previous surgeries which could have played a role in the unilateral expression of BMS. In many cases the patient remains asymptomatic although persistent pain may appear due to bullous keratopathy. This pain is related to the rupture of the bullae and the ensuing exposure of corneal nervous endings. If treatment with hypertonic saline solution or lubricants is not enough, extended use soft contact lenses may be required with the ensuing risk of infection. In the case of progressive decompensation which could affect the center of the cornea, as can occur particularly with anterior chamber phakic lenses, penetrating keratoplasty could be indicated. However, this would not be adequate in
a r c h s o c e s p o f t a l m o l . 2 0 1 3;8 8(5):193–196
195
Fig. 3 – (A) Two weeks after the puncture, without contact lens and (B) subepithelial fibrosis in the punctured area after 3 weeks. Fig. 2 – (A) Stromal micropuncture with cystotome at about 150 m depth and (B) fluorescein image immediately after the puncture.
the typical syndrome because the VA is maintained and larger diameters would be required, thus significantly increasing the risk of surgery due to rejection caused by proximity to the limbus. The possibility of carrying out an “annular” Descemet Stripping Automated Endothelial Keratoplasty (DSAEK) would not be feasible. The patient of this report exhibited a spontaneous and highly satisfactory VA of 20/30, for which reason keratoplasty of any type was discarded. Syndrome cases with association to anterior chamber phakic lenses in which the condition has been halted by withdrawing the lens are altogether different. Stromal puncture has been applied as an alternative to keratoplasty in eyes with bullous keratoplasty without visual expectations and in cases of relapsing erosion. The mechanisms by means of which ASM relieves discomfort in symptomatic patients will therefore be related to the disappearance of epithelial bullae although the stroma exhibits edema and thickening due to endothelial pump failure. The biomicroscopic finding of subepithelial fibrosis after ASM can be explained at the histological level with the appearance of epithelial–stromal projections which would act as anchoring system of the epithelium to the base membrane, preventing the penetration of liquid in the subepithelial space.5
It has been suggested that stromal puncture may activate the basal cells layer and keratocytes, thus stimulating the expression of extracellular matrix proteins (fibronectin, laminin, collagen type IV). In 2004, Martins3 successfully performed ASM in 2 symptomatic BMS cases, both aphakic. Micropuncture was performed with 25 G needle with bent tip. It was necessary to repeat the procedure in one case. The instant case is pseudophakic without evidencing pseudophakodonesis. The procedure was performed with a disposable cystotome that was used for phacoemulsification surgery as its curvature facilitated puncturing. ASM reduces peripheral edema and alleviates pain in BMS symptomatic patients, being a simple and low cost procedure.
Conflict of interests No conflict of interests has been declared by the authors.
references
1. Brown SI, McLean JM. Peripheral corneal edema after cataract extraction: a new clinical entity. Trans Am Acad Ophthalmol Otolaryngol. 1969;73:465–70.
196
a r c h s o c e s p o f t a l m o l . 2 0 1 3;8 8(5):193–196
2. Cormier G, Brunette I, Boisjoly HM, LeFranc¸ois M, Shi ZH, Guertin MC. Anterior stromal punctures for bullous keratopathy. Arch Ophthalmol. 1996;114:654–8. 3. Gothard TW, Hardten DR, Lane SS, Doughman DJ, Krachmer JH, Holland EJ. Clinical findings in Brown–McLean syndrome. Am J Ophthalmol. 1993;115:729–37.
4. Martins EN, Alvarenga LS, Sousa LB, Orlando Filho VT, Gomes JA, De Freitas D. Anterior stromal puncture in Brown–McLean syndrome. J Cataract Refract Surg. 2004;30:1575–7. 5. Hsu JK, Rubinfeld RS, Barry P, Jester JV. Anterior stromal puncture. Immunohistochemical studies in human corneas. Arch Ophthalmol. 1993;111:1057–63.
ARCHIVOS DE LA SOCIEDAD ESPAÑOLA DE OFTALMOLOGÍA www.elsevier.es/oftalmologia
Short communication
Anterior stromal puncture for the treatment of Brown–McLean syndrome夽,夽夽 P. Rodríguez-Ausín ∗ , K. Pachkoria Clínica Oftalmológica Suárez Leoz, Madrid, Spain
a r t i c l e
i n f o
a b s t r a c t
Article history:
Case report: The Brown–McLean syndrome is defined by peripheral corneal edema sparing
Received 18 December 2011
the central cornea. We report a patient with bullous annular keratopathy following pha-
Accepted 8 April 2012
coemulsification and implantation of posterior chamber intraocular lens. The uncorrected
Available online 28 June 2013
and best corrected visual acuity was 20/30. The patient required the use of a bandage contact lens for almost 5 years to relieve the ocular discomfort.
Keywords:
Discussion: Anterior stromal puncture was performed using a cystotome. Two weeks after
Peripheral corneal edema
the procedure the ocular discomfort disappeared and the use of bandage contact lens was
Anterior stromal puncture
no longer required. Anterior stromal puncture is a simple procedure that is performed under
Bullous keratopathy
the slit lamp, and can alleviate the symptoms in patients with Brown–McLean syndrome.
Brown–McLean syndrome
© 2011 Sociedad Española de Oftalmología. Published by Elsevier España, S.L. All rights reserved.
Keratoplasty
Micropunción estromal anterior como tratamiento del síndrome de Brown–McLean r e s u m e n Palabras clave:
El síndrome Brown–McLean consiste en la presencia de edema corneal periférico respetando
Edema corneal periférico
˜ la córnea central. Presentamos un paciente con queratopatia bullosa anular anos después
Punción estromal anterior
de facoemulsificación e implante de lente en cámara posterior. Su agudeza visual espon-
Queratopatía bullosa
tánea y corregida era 20/30 y precisaba lente de contacto blanda para aliviar molestias desde
Síndrome de Brown–McLean
˜ hacía 5 anos. Practicamos micropunción estromal anterior con cistitomo, y en 2 semanas
Queratoplastia
las molestias desaparecieron y no precisa porte de lente blanda. La micropunción estromal es un procedimiento simple que se realiza en lámpara de hendidura y que puede eliminar la sintomatología del síndrome de Brown–McLean. © 2011 Sociedad Española de Oftalmología. Publicado por Elsevier España, S.L. Todos los derechos reservados.
夽 Please cite this article as: Rodríguez-Asuín P, Pachkoria K. Micropunción estromal anterior como tratamiento del síndrome de Brown–McLean. Arch Soc Esp Oftalmol. 2013;88:193–6. 夽夽 ˜ Presented as a verbal communication at the 87.◦ Congreso de la Sociedad Espanola de Oftalmología, Oviedo, 2011. ∗ Corresponding author. E-mail address: [email protected] (P. Rodríguez-Ausín).
2173-5794/$ – see front matter © 2011 Sociedad Española de Oftalmología. Published by Elsevier España, S.L. All rights reserved.
194
a r c h s o c e s p o f t a l m o l . 2 0 1 3;8 8(5):193–196
Introduction The Brown–McLean syndrome (BMS) consists in the appearance of peripheral corneal edema sparing the central area. It was first described in 19691 and is generally associated to previous intracapsular lens surgery. BMS patients are usually asymptomatic although some documented cases exhibited chronic irritation symptoms, pain and septic corneal ulcers. Various treatment options with varying degrees of success have been published for symptomatic patients including hypertonic saline solutions or lubricants,2 extended use soft contact lenses, penetrating keratoplasty, removal of the phakic lens in the anterior chamber and anterior stromal micropuncture.3 This paper presents a symptomatic BMS case successfully treated with anterior stromal micropuncture (ASM).
Case report Male patient, 72, intervened for phacoemulsification with intraocular lens implant in both eyes 12 years before, also requiring vitrectomy for removing nuclear remains in the right eye. The patient had to use soft contact lenses starting 5 years before with substitution every 2 months due to persistent discomfort in the right eye (RE). Non-corrected and best corrected visual acuity was of 20/30 in both eyes (BE). Biomicroscopy revealed pseudophakia in the anterior chamber of BE and a 3 mm peripheral annular corneal edema in the RE sparing the central cornea with peripheral endothelial pigment dispersion (Fig. 1A and B). No gutattas were observed in BE. The LE cornea did not exhibit pathological findings. Intraocular pressure (IOP) was normal in BE and central endothelial cell density measured with SP-3000P TOPCON® mirror microscope was of 1300 and 1500 cells/mm2 in RE and LE respectively. The peripheral density was not displayed due to edema in RE. In April 2011, ASM was performed in slit lamp under topical anesthesia with 0.5% tetracaine (Colircusí Anestésico® , Alcon-Cusí, Barcelona, Spain), consisting in 40–50 superficial micropunctures (one fourth thickness, approximately 150 m) in each quadrant utilizing a 25 G cystotome (BD VisitecTM , Waltham, USA) sparing the central cornea (Fig. 2A and B). Antibiotic prophylaxis was applied with moxifloxacin (Vigamox® , Alcon-Cusí, El Masnou, Barcelona, Spain) before and after the procedure. 2 weeks later, the patient was able to stop using the contact lens undisturbed and in 3 weeks subepithelial fibrosis was observed in the treated area. The patient remains asymptomatic after 12 months of follow-up (Fig. 3A and B).
Discussion BMS consists in peripheral corneal edema with circumferential progression which typically spares the central cornea. In addition, dotted brownish-orange pigmentation may appear in the peripheral endothelium. Central endothelial density is somewhat below normal values and cornea gutatta may coexist. It has been described mainly after intracapsular lens extraction and with less frequency after extracapsular
Fig. 1 – (A) Cornea with blurred ring and presence of soft contact lens and (B) peripheral edema with pigmented deposits.
extraction, phacoemulsification, keratoplasty, IOL implant in CA and vitrectomy. In addition, it has been reported in the absence of surgery associated to ocular traumatism, closed angle glaucoma, myotonic dystrophy and lens subluxation.3,4 Suggested pathogenics include iris peripheral endothelial traumatism, IOL haptics, dislocated vitreous or lens which causes endothelial abrasion and cell loss in the periphery, without discarding associated genetic predisposition.4 The period of time in which BMS appears after surgery has been reported between 1.5 and 34 years. In the instant case it appeared after 10 years and unilaterally. This extended period of time has made us consider progressive RE endothelial damage without knowing the intra- and post surgery details of previous surgeries which could have played a role in the unilateral expression of BMS. In many cases the patient remains asymptomatic although persistent pain may appear due to bullous keratopathy. This pain is related to the rupture of the bullae and the ensuing exposure of corneal nervous endings. If treatment with hypertonic saline solution or lubricants is not enough, extended use soft contact lenses may be required with the ensuing risk of infection. In the case of progressive decompensation which could affect the center of the cornea, as can occur particularly with anterior chamber phakic lenses, penetrating keratoplasty could be indicated. However, this would not be adequate in
a r c h s o c e s p o f t a l m o l . 2 0 1 3;8 8(5):193–196
195
Fig. 3 – (A) Two weeks after the puncture, without contact lens and (B) subepithelial fibrosis in the punctured area after 3 weeks. Fig. 2 – (A) Stromal micropuncture with cystotome at about 150 m depth and (B) fluorescein image immediately after the puncture.
the typical syndrome because the VA is maintained and larger diameters would be required, thus significantly increasing the risk of surgery due to rejection caused by proximity to the limbus. The possibility of carrying out an “annular” Descemet Stripping Automated Endothelial Keratoplasty (DSAEK) would not be feasible. The patient of this report exhibited a spontaneous and highly satisfactory VA of 20/30, for which reason keratoplasty of any type was discarded. Syndrome cases with association to anterior chamber phakic lenses in which the condition has been halted by withdrawing the lens are altogether different. Stromal puncture has been applied as an alternative to keratoplasty in eyes with bullous keratoplasty without visual expectations and in cases of relapsing erosion. The mechanisms by means of which ASM relieves discomfort in symptomatic patients will therefore be related to the disappearance of epithelial bullae although the stroma exhibits edema and thickening due to endothelial pump failure. The biomicroscopic finding of subepithelial fibrosis after ASM can be explained at the histological level with the appearance of epithelial–stromal projections which would act as anchoring system of the epithelium to the base membrane, preventing the penetration of liquid in the subepithelial space.5
It has been suggested that stromal puncture may activate the basal cells layer and keratocytes, thus stimulating the expression of extracellular matrix proteins (fibronectin, laminin, collagen type IV). In 2004, Martins3 successfully performed ASM in 2 symptomatic BMS cases, both aphakic. Micropuncture was performed with 25 G needle with bent tip. It was necessary to repeat the procedure in one case. The instant case is pseudophakic without evidencing pseudophakodonesis. The procedure was performed with a disposable cystotome that was used for phacoemulsification surgery as its curvature facilitated puncturing. ASM reduces peripheral edema and alleviates pain in BMS symptomatic patients, being a simple and low cost procedure.
Conflict of interests No conflict of interests has been declared by the authors.
references
1. Brown SI, McLean JM. Peripheral corneal edema after cataract extraction: a new clinical entity. Trans Am Acad Ophthalmol Otolaryngol. 1969;73:465–70.
196
a r c h s o c e s p o f t a l m o l . 2 0 1 3;8 8(5):193–196
2. Cormier G, Brunette I, Boisjoly HM, LeFranc¸ois M, Shi ZH, Guertin MC. Anterior stromal punctures for bullous keratopathy. Arch Ophthalmol. 1996;114:654–8. 3. Gothard TW, Hardten DR, Lane SS, Doughman DJ, Krachmer JH, Holland EJ. Clinical findings in Brown–McLean syndrome. Am J Ophthalmol. 1993;115:729–37.
4. Martins EN, Alvarenga LS, Sousa LB, Orlando Filho VT, Gomes JA, De Freitas D. Anterior stromal puncture in Brown–McLean syndrome. J Cataract Refract Surg. 2004;30:1575–7. 5. Hsu JK, Rubinfeld RS, Barry P, Jester JV. Anterior stromal puncture. Immunohistochemical studies in human corneas. Arch Ophthalmol. 1993;111:1057–63.