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Spiroplasma infection as a cause of severe congenital keratouveitis, cataract and glaucoma

Helena Van Haecke, Dimitri Roels (UGent) , Fanny Nerinckx (UGent) , Heidi Schaballie (UGent) , Petra Schelstraete (UGent) , Linos Vandekerckhove (UGent) , Jolien Van Cleemput (UGent) , Wim Van Den Broeck (UGent) , Liesbeth Couck (UGent) , Hannelore Hamerlinck (UGent) , et al.
(2024) BMC OPHTHALMOLOGY. 24(1).
Author
Organization
Abstract
Background Only seven cases of ocular Spiroplasma infection have been reported to date, all presenting as congenital cataracts with concomitant intraocular inflammation. We describe the first case of Spiroplasma infection initially presenting as a corneal infiltrate. Case presentation A 1-month-old girl was referred for a corneal infiltrate in the left eye. She presented in our hospital with unilateral keratouveitis. Examination showed a stromal corneal infiltrate and dense white keratic precipitates in the left eye. Herpetic keratouveitis was suspected and intravenous acyclovir therapy was initiated. Two weeks later, the inflammation in the left eye persisted and was also noticed in the right eye. Acute angle-closure glaucoma and a cataract with dilated iris vessels extending onto the anterior lens capsule developed in the left eye. The inflammation resolved after treatment with azithromycin. Iridectomy, synechiolysis and lensectomy were performed. Bacterial metagenomic sequencing (16 S rRNA) and transmission electron microscopy revealed Spiroplasma ixodetis species in lens aspirates and biopsy. Consequently, a diagnosis of bilateral Spiroplasma uveitis was made. Conclusions In cases of congenital cataract with concomitant intraocular inflammation, Spiroplasma infection should be considered. The purpose of this case report is to raise awareness of congenital Spiroplasma infection as a cause of severe keratouveitis, cataract and angle-closure glaucoma in newborns. Performing molecular testing on lens aspirates is essential to confirm diagnosis. Systemic macrolides are suggested as the mainstay of treatment.
Keywords
Congenital keratouveitis, Cataract, Angle-closure glaucoma, Spiroplasma ixodetis

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MLA
Van Haecke, Helena, et al. “Spiroplasma Infection as a Cause of Severe Congenital Keratouveitis, Cataract and Glaucoma.” BMC OPHTHALMOLOGY, vol. 24, no. 1, 2024, doi:10.1186/s12886-024-03480-z.
APA
Van Haecke, H., Roels, D., Nerinckx, F., Schaballie, H., Schelstraete, P., Vandekerckhove, L., … Joniau, I. (2024). Spiroplasma infection as a cause of severe congenital keratouveitis, cataract and glaucoma. BMC OPHTHALMOLOGY, 24(1). https://doi.org/10.1186/s12886-024-03480-z
Chicago author-date
Van Haecke, Helena, Dimitri Roels, Fanny Nerinckx, Heidi Schaballie, Petra Schelstraete, Linos Vandekerckhove, Jolien Van Cleemput, et al. 2024. “Spiroplasma Infection as a Cause of Severe Congenital Keratouveitis, Cataract and Glaucoma.” BMC OPHTHALMOLOGY 24 (1). https://doi.org/10.1186/s12886-024-03480-z.
Chicago author-date (all authors)
Van Haecke, Helena, Dimitri Roels, Fanny Nerinckx, Heidi Schaballie, Petra Schelstraete, Linos Vandekerckhove, Jolien Van Cleemput, Wim Van Den Broeck, Liesbeth Couck, Hannelore Hamerlinck, Stien Vandendriessche, Jerina Boelens, and Inge Joniau. 2024. “Spiroplasma Infection as a Cause of Severe Congenital Keratouveitis, Cataract and Glaucoma.” BMC OPHTHALMOLOGY 24 (1). doi:10.1186/s12886-024-03480-z.
Vancouver
1.
Van Haecke H, Roels D, Nerinckx F, Schaballie H, Schelstraete P, Vandekerckhove L, et al. Spiroplasma infection as a cause of severe congenital keratouveitis, cataract and glaucoma. BMC OPHTHALMOLOGY. 2024;24(1).
IEEE
[1]
H. Van Haecke et al., “Spiroplasma infection as a cause of severe congenital keratouveitis, cataract and glaucoma,” BMC OPHTHALMOLOGY, vol. 24, no. 1, 2024.
@article{01J1SNCBGFXT5T14PH83HA1F2S,
  abstract     = {{Background Only seven cases of ocular Spiroplasma infection have been reported to date, all presenting as congenital cataracts with concomitant intraocular inflammation. We describe the first case of Spiroplasma infection initially presenting as a corneal infiltrate. Case presentation A 1-month-old girl was referred for a corneal infiltrate in the left eye. She presented in our hospital with unilateral keratouveitis. Examination showed a stromal corneal infiltrate and dense white keratic precipitates in the left eye. Herpetic keratouveitis was suspected and intravenous acyclovir therapy was initiated. Two weeks later, the inflammation in the left eye persisted and was also noticed in the right eye. Acute angle-closure glaucoma and a cataract with dilated iris vessels extending onto the anterior lens capsule developed in the left eye. The inflammation resolved after treatment with azithromycin. Iridectomy, synechiolysis and lensectomy were performed. Bacterial metagenomic sequencing (16 S rRNA) and transmission electron microscopy revealed Spiroplasma ixodetis species in lens aspirates and biopsy. Consequently, a diagnosis of bilateral Spiroplasma uveitis was made. Conclusions In cases of congenital cataract with concomitant intraocular inflammation, Spiroplasma infection should be considered. The purpose of this case report is to raise awareness of congenital Spiroplasma infection as a cause of severe keratouveitis, cataract and angle-closure glaucoma in newborns. Performing molecular testing on lens aspirates is essential to confirm diagnosis. Systemic macrolides are suggested as the mainstay of treatment.}},
  articleno    = {{217}},
  author       = {{Van Haecke, Helena and Roels, Dimitri and Nerinckx, Fanny and Schaballie, Heidi and Schelstraete, Petra and Vandekerckhove, Linos and Van Cleemput, Jolien and Van Den Broeck, Wim and Couck, Liesbeth and Hamerlinck, Hannelore and Vandendriessche, Stien and Boelens, Jerina and Joniau, Inge}},
  issn         = {{1471-2415}},
  journal      = {{BMC OPHTHALMOLOGY}},
  keywords     = {{Congenital keratouveitis,Cataract,Angle-closure glaucoma,Spiroplasma ixodetis}},
  language     = {{eng}},
  number       = {{1}},
  pages        = {{5}},
  title        = {{Spiroplasma infection as a cause of severe congenital keratouveitis, cataract and glaucoma}},
  url          = {{http://doi.org/10.1186/s12886-024-03480-z}},
  volume       = {{24}},
  year         = {{2024}},
}
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