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Deep lamellar keratoplasty in corneal dermoid

Authors:
Ritu Arora at Guru Nanak Eye Centre
Ritu Arora
V Jain
V Jain
V Jain
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D Mehta
D Mehta
D Mehta
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Pasterova 2, 11000 Beograd
Serbia and Montenegro
Correspondence: S Milenkovic
´
Tel: þ381 11 688 997
Fax: þ381 11 2688 164
E-mail: milenkov@net.yu
Eye (2005) 19, 917–920. doi:10.1038/sj.eye.6701670;
published online 10 September 2004
Sir,
Deep lamellar keratoplasty in corneal dermoid
Corneal dermoids are a rare cause of corneal
opacification consisting of abnormal mesoblastic tissue
covered by the epithelium. They can involve the deeper
layers of cornea leaving an intact descemet’s membrane
and endothelium. Occasionally, anterior segment
structures may be involved by the choristomatous
growth and there may occur accompanying ocular
malformations. A surgical intervention in the form of
lamellar keratoplasty (LK) or penetrating keratoplasty
(PK) is warranted on accounts of cosmesis, discomfort,
and primarily because they are located in the visual axis.
Recently, we treated a child with a corneal dermoid
encroaching on to the visual axis. An anterior deep
lamellar keratoplasty was performed after the
assessment of depth of involvement of the corneal mass
by ultrasound biomicroscopy. To our knowledge this is
the first report of a successful deep LK in a patient with a
corneal dermoid.
Case report
An 8-year-old boy presented with the complaints of
pinkish mass and decreased vision in his left eye. This
mass was present since birth and had gradually
increased in size. The child was the product of normal
uncomplicated gestation and had normal development.
Ocular examination disclosed a normal right eye with
a visual acuity of 6/6. Left-eye visual acuity was hand
movements close to face. Constant divergent squint of
approximately 30-prism diopter was present in the left
eye. Eyelids were normal in both eyes. Left eye showed a
vascularized, moderately elevated, sharply
circumscribed pinkish mass 6 6mm
2
in size overlying
the cornea superonasally. Also present was a ring of lipid
deposit around the mass in the adjacent clear cornea
(Figure 1a). The remaining cornea was clear. Anterior
segment was otherwise normal. Clinically, a diagnosis of
corneal dermoid was made. Ultrasound biomicroscopy
(UBM) disclosed a highly echogenic lesion occupying the
superficial 60% of the cornea. Deeper part of stroma,
descemet’s membrane and endothelium were not
involved (Figure 2a). An anterior deep LK (DLK) was
planned.
After a partial thickness corneal trephination with
6 mm trephine, a superficial keratectomy involving the
mass was performed. A disposable 30-gauge needle was
inserted deeply with bevel down into the paracentral
stroma and air was injected. A small opening was made
in the air bubble and the remaining stromal layers were
removed till the descemet’s membrane was bared of
the stroma. Donor corneal button 6.5 mm sized, stripped
of descemet’s membrane was sutured onto the bare
descemet’s membrane. In all, 16 interrupted 10/0 nylon
sutures were given. Postoperatively, the eye was
treated with topical prednisolone acetate (1%),
tobramycin (0.3%) and a tear substitute four times
a day. Histopathology of the corneal dermoid revealed
thick keratinized epithelium and sebaceous glands
enmeshed in connective tissue (Figure 2b). Patient
had an uneventful postoperative course. Visual acuity
improved to 3/60 and is currently receiving ambylopia
therapy. At 6 months postoperatively the graft has
remained transparent with no interface scarring
(Figure 1b).
Figure 1 (a) Clinical photograph of left eye showing smooth,
elevated, sharply circumscribed mass with an arc of lipid in
adjacent cornea. (b) Left eye 6 months after DLK showing clear
graft.
Figure 2 (a) Ultrasound biomicroscopic picture showing highly
echogenic lesion occupying the superficial 60% of the cornea. (b)
Histopathology slide showing thick keratinized epithelium and
a sebaceous gland enmeshed in connective tissue (H&E, 25).
Correspondence
920
Eye
Comment
Corneal dermoid is a congenital benign tumour
consisting of tissues of ectodermal and mesodermal
origin appearing as raised yellowish white vascularized
bulbous lesions.
1
Mann
2
classified corneal dermoids into three broad
types. Our case belonged to grade II with corneal
involvement sparing deep stroma, descemet’s
membrane, and endothelium. This was elucidated well
on UBM.
The surgical management of corneal dermoid depends
on the size, site and depth of involvement.
3
Simple
excision is generally not sufficient by itself to manage an
extensive lesion.
4
Corneal dermoids with no anterior chamber
involvement require LK in which the lesion is excised to
its entirety and a lamellar graft is tailored to fit the defect.
1
LK has the advantage of avoiding most postoperative
complications associated with PK especially less risk of
allograft rejection but has the major disadvantage of
interface scarring and hazy graft. Of late, DLK is being
performed more commonly over LK/PK with minimal
reports of interface scarring. We decided to perform DK
using big bubble technique as UBM elucidated the lesion
to be distinct and sparing the underlying descemet’s
membrane and endothelium.
The case is being reported because of the rarity of the
condition and use of relatively new diagnostic tool UBM
to assist in its management. To the best of our knowledge
DLK has not been previously reported in corneal
dermoid management.
References
1 Henkind P, Marinoff G, Manas A, Friedman A. Bilateral
corneal dermoids. Am J Ophthalmol 1973; 76: 971–977.
2 Mann I. Development Abnormalities of the Eye. Lippincott:
Philadelphia, 1957, pp 357–364.
3 Golubovic S, LatKovic Z, Horyatic OM. Surgical treatment of
larger corneal dermoid. Doc Ophthalmol 1995; 91: 25–32.
4 Mohan M, Mukherjee G, Panda A. Clinical evaluation and
surgical intervention of limbal dermoid. Indian J Ophthalmol
1981; 29: 69–73.
R Arora, V Jain and D Mehta
Cornea Services, Guru Nanak Eye Center
New Delhi 110002, India
Correspondence: R Arora
D-1, Nizammudin
West New Delhi 110013, India
Tel: þ91 1124351415
E-mail: aroraj@del3.vsnl.net.in
Eye (2005) 19, 920–921. doi:10.1038/sj.eye.6701672;
published online 3 September 2004
Sir,
Stenotrophomonas maltophilia keratitis after
penetrating keratoplasty
Stenotrophomonas maltophilia is an aerobic, Gram-negative
ubiquitous bacillus, isolated from water, soil, plants,
and animals.
1
Previously described to be of limited
pathogenic potential, it is now emerging as
an important nosocomial pathogen.
2
Microbial
keratitis due to S. maltophilia is rare with only 11
cases reported in literature.
3–7
We report a case of
S. maltophilia keratitis following penetrating keratoplasty
that was managed by topical fluoroquinolone
monotherapy.
Case report
A 70-year-old lady presented with diminished vision of
1 week’s duration in her right eye. In the affected eye a
penetrating keratoplasty was done for a corneal scar
(a sequel of burnt-out trachoma) 5.5 months earlier.
Postoperatively, a persistent epithelial defect resolved
over 2 months with topical preservative-free tear
substitutes, antibiotic eyedrops and a bandage contact
lens. On examination, her best-corrected visual
acuity in the right eye was 20/400. She had
lagophthalmos with no corneal exposure. The lid
margins were thickened and irregular with significant
meibomitis. Trichiasis was not noted. The graft–host
junction was well-apposed. There was no bandage
contact lens. There was a central epithelial defect with
an underlying stromal infiltrate (3.5 3.7 mm) and
surrounding stromal oedema (Figure 1a). The remaining
details were not visualised. An ultrasound B scan
of the right eye was normal. Grams, Giemsa, and
KOH stains of the corneal scrapings revealed no
organisms. She was started on half-hourly fortified
Cefazolin eyedrops (50 mg/ml) and fortified Gentamicin
eyedrops (14 mg/ml). These were discontinued after 2
days due to significant growth of Gram-negative bacilli
in culture, sensitive only to ciprofloxacin and
chloramphenicol. The organism was identified as
Stenotrophomonas maltophilia by API 20NE (API,
Biomerieux, France). Gradual resolution with 0.3%
ciprofloxacin hydrochloride eyedrops was noted. A
measure of 0.1% betamethasone sulphate was added to
reduce inflammation. The lesion healed after 2.5 months
of therapy (Figure 1b). She is currently awaiting a regraft
in the right eye.
Correspondence
921
Eye
... S. maltophilia, which is usually free living in the environment has been implicated in nosocomial infections and community based infections (Köseoglu et al., 2004;Meyer et al., 2006;Falagas et al., 2009). It has been reported as etiological agents in bacteraemia, ocular infection, endocarditis and RTIs (associated Labarca et al., 2000;Friedman et al., 2002;Senol et al., 2002;Al-Anazi et al., 2006;Jaidane et al., 2014 Water andwastewater Municipal Chang et al., 2005;Adjidé et al., 2010 Tropical pyomyositis Thomas et al., 2010 Cystic fibrosis Talmaciu et al., 2000;Di Bonaventura et al., 2007;Hansen, 2012Microfiltered water dispensers Sacchetti et al., 2009River water Nakatsu et al., 1995 Intestinal colonization resulting in diarrhea Apisarnthanarak et al., 2003 Saline subterranean Lake Rivas et al., 2009 Septic arthritis Aydemir et al., 2008Showerheads Feazel et al., 2009Drinking water Simões et al., 2007Silbaq, 2009Endocarditis Takigawa et al., 2008 with cystic fibrosis), wound infection and urinary tract infections (UTI) (Kim et al., 2002;Platsouka et al., 2002;Arora et al., 2005). It is also an aetiologic agents of meningitis, sepsis, skin, and soft tissue infections (SSTI) and it has been diagnosed with rare cases of pyomyositis (Gales et al., 2001;Platsouka et al., 2002;Sakhnini et al., 2002;Arora et al., 2005;Pathmanathan and Waterer, 2005;Al-Anazi et al., 2006;Yemisen et al., 2008;Thomas et al., 2010). ...
... It has been reported as etiological agents in bacteraemia, ocular infection, endocarditis and RTIs (associated Labarca et al., 2000;Friedman et al., 2002;Senol et al., 2002;Al-Anazi et al., 2006;Jaidane et al., 2014 Water andwastewater Municipal Chang et al., 2005;Adjidé et al., 2010 Tropical pyomyositis Thomas et al., 2010 Cystic fibrosis Talmaciu et al., 2000;Di Bonaventura et al., 2007;Hansen, 2012Microfiltered water dispensers Sacchetti et al., 2009River water Nakatsu et al., 1995 Intestinal colonization resulting in diarrhea Apisarnthanarak et al., 2003 Saline subterranean Lake Rivas et al., 2009 Septic arthritis Aydemir et al., 2008Showerheads Feazel et al., 2009Drinking water Simões et al., 2007Silbaq, 2009Endocarditis Takigawa et al., 2008 with cystic fibrosis), wound infection and urinary tract infections (UTI) (Kim et al., 2002;Platsouka et al., 2002;Arora et al., 2005). It is also an aetiologic agents of meningitis, sepsis, skin, and soft tissue infections (SSTI) and it has been diagnosed with rare cases of pyomyositis (Gales et al., 2001;Platsouka et al., 2002;Sakhnini et al., 2002;Arora et al., 2005;Pathmanathan and Waterer, 2005;Al-Anazi et al., 2006;Yemisen et al., 2008;Thomas et al., 2010). Clinical skin presentations include primary cellulitis, cellulitis-like cutaneous metastasis or cellulitis or metastatic nodular skin lesions, gangrenous cellulitis, ecthyma gangrenosum, soft-tissue necrosis, and infected mucocutaneous ulcers (Denton and Kerr, 1998;Foo et al., 2002;Teo et al., 2006;Smeets et al., 2007). ...
Stenotrophomonas maltophilia as an Emerging Ubiquitous Pathogen: Looking Beyond Contemporary Antibiotic Therapy
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  • Nov 2017
Stenotrophomonas maltophilia is a commensal and an emerging pathogen earlier noted in broad-spectrum life threatening infections among the vulnerable, but more recently as a pathogen in immunocompetent individuals. The bacteria are consistently being implicated in necrotizing otitis, cutaneous infections including soft tissue infection and keratitis, endocarditis, meningitis, acute respiratory tract infection (RTI), bacteraemia (with/without hematological malignancies), tropical pyomyositis, cystic fibrosis, septic arthritis, among others. S. maltophilia is also an environmental bacteria occurring in water, rhizospheres, as part of the animals' microflora, in foods, and several other microbiota. This review highlights clinical reports on S. maltophilia both as an opportunistic and as true pathogen. Also, biofilm formation as well as quorum sensing, extracellular enzymes, flagella, pili/fimbriae, small colony variant, other virulence or virulence-associated factors, the antibiotic resistance factors, and their implications are considered. Low outer membrane permeability, natural MDR efflux systems, and/or resistance genes, resistance mechanisms like the production of two inducible chromosomally encoded β-lactamases, and lack of carefully compiled patient history are factors that pose great challenges to the S. maltophilia control arsenals. The fluoroquinolone, some tetracycline derivatives and trimethoprim-sulphamethaxole (TMP-SMX) were reported as effective antibiotics with good therapeutic outcome. However, TMP-SMX resistance and allergies to sulfa together with high toxicity of fluoroquinolone are notable setbacks. S. maltophilia's production and sustenance of biofilm by quorum sensing enhance their virulence, resistance to antibiotics and gene transfer, making quorum quenching an imperative step in Stenotrophomonas control. Incorporating several other proven approaches like bioengineered bacteriophage therapy, Epigallocatechin-3-gallate (EGCG), essential oil, nanoemulsions, and use of cationic compounds are promising alternatives which can be incorporated in Stenotrophomonas control arsenal.
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... During the examination, signs of clinical keratoconjunctivitis and anterior uveitis were recorded, if present. Corneal lesions assessed by biomicroscopy were assigned injury scores similar to Arora's classification (Arora et al., 2005). Further details regarding the eye examination techniques can be found in Olanrewaju et al. (2016). ...
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... During the examination, signs of clinical keratoconjunctivitis and anterior uveitis were recorded, if present. Corneal lesions assessed by biomicroscopy were assigned injury scores similar to Arora's classification (Arora et al., 2005). Details regarding the eye examination techniques can be found in Olanrewaju et al. (2016). ...
Influence of light sources and photoperiod on growth performance, carcass characteristics, and health indices of broilers grown to heavy weights2
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... DLKP can be performed in almost all cases of corneal opacity that have a healthy endothelial cell count. It has been reported for Keratoconus (Coombes et al., 2001;Shimmura et al., 2005;Al-Torbak et al., 2006) corneal stromal dystrophies (Shimmura and Tsubota, 2006;Kawashima et al., 2006), corneal leucoma (Sugita and Kondo, 1997;Senoo et al., 2005), corneal dermoid (Arora et al., 2005), infectious corneal opacity (Sugita and Kondo, 1997;Senoo et al., 2005) and sever ocular surface disease (Yao et al., 2002;Fogla and Padmanabhan, 2005). Corneal perforations after infection or immunologic disease can be treated by therapeutic DLKP with satisfactory result (Shimmura et al., 2003;Tong et al., 2004). ...
Deep anterior lamellar Keratoplasty
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Introduction Advanced surgical techniques, better post-operative protocols, imaging, and better understanding of genetic basis have enhanced outcomes of pediatric keratoplasty. However, results in infants and younger children remain a challenge. Transplants in the pediatric age group are challenging because of the complexity of the indications, the procedure itself, and the problems with respect to follow-up and post-operative care in younger recipients. Areas covered This review presents an overview of challenges faced in the management of pediatric corneal grafts, and problems encountered in long-term survival. We discuss the changing trends in these outcomes of PKP with a current review of the recent literature from PubMed. We also share the results of pediatric keratoplasty done at our center in the last three years and have an in-depth discussion about the management of comorbidities like cataracts and glaucoma Expert opinion Despite several advances in microsurgical techniques for corneal transplantation, pediatric keratoplasty remains challenging due to a variety of factors such as young age, repeated need for anesthesia, immature ocular tissues, and accelerated healing. The advent of component surgeries like DALK, DSEK, and DMEK has improved outcomes and simplified management options. Even after specialized surgeries, long-term follow-ups and management of comorbidities become essential.
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Large Congenital Corneal Dermoid With Spontaneous Partial Regression: The First Report
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To report a spontaneous partial regression of a large congenital corneal dermoid in a newborn. Review and follow-up of the medical records of a female newborn presenting with a tumor mass of her right eye and no other congenital abnormalities. The child presented on the first day of life with a mushroom-shaped mass of 15 × 15 mm in diameter that involved the most of the cornea leaving a small clear portion superiorly. The mass protruded through the palpebral aperture. A diagnosis of corneal dermoid was made based on clinical presentation of the mass and radiographic and ultrasound findings. Enucleation of the right eye was discussed, but declined by parents. During the first month of life, a spontaneous partial regression of the mass occurred, so that the patient was able to close the lids completely above the mass. This dermoid is the third type according to Mann's classification. Characterized by involvement of the entire anterior segment and lack of lens. To the best of our knowledge, the case reported is the first documented spontaneous partial regression of a large corneal dermoid. Surgical excision at an early age may be recommended to avoid development of amblyopia and dramatic growth of the tumor. In our case the tumor regressed and surgical excision would have no impact on visual outcome. Future management of type III corneal dermoid needs to consider whether to perform surgery at an early stage or to recommend conservative management.
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Bilateral Corneal Dermoids
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  • Jan 1974
  • AM J OPHTHALMOL
Two male cousins of Puerto Rican extraction were born with bilateral opaque corneas, but no other ocular or systemic abnormalities. Family history revealed that the infants were born of sisters with unrelated husbands. Histopathologic examination revealed that the opacifications were due to corneal dermoids that involved almost the entire corneal surface but spared the limbus. The dermoids occupied only the anterior portion of the cornea. In the more severely affected infant, a penetrating corneal graft failed, but a lamellar graft in the opposite eye succeeded. The condition of total corneal dermoid must be distinguished from many other causes of neonatal corneal opacification. It may be confused with congenital hereditary corneal dystrophy or sclerocornea. Corneal biopsy may be useful in making the diagnosis in cases of neonatal corneal opacification. These cases seemed to be the first recorded human instances of hereditary corneal dermoids.
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Surgical treatment of large corneal dermoid
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  • Feb 1995
Corneal and limbal dermoids are uncommon choristomatous corneal tumors. They clinically present as round or oval, whitish or yellowish cones protruding on the anterior surface of the eyeball. They are composed of ectodermal (keratinized epithelium, hairs, sebaceous and sudoriferous glands, nerves, smooth muscles and, less frequently, teeth) and mesodermal elements (fibrous tissue, fat, blood vessels and cartilage) combined in different proportion. If fat dominates in histology of the tumor, it is called a lipodermoid. A case of a two-year old boy with a large corneal dermoid on the right eye is presented. Dermoid covered almost the whole cornea and was associated with adherence of the atrophic iris to the posterior corneal surface and partial congenital cataract. Surgical therapy included excision of dermoid together with the superficial lamellae of the corneal stroma and penetrating keratoplasty. Satisfactory tectonic and esthetic outcome was achieved and has been maintained for four postoperative years now.
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Development Abnormalities of the Eye
  • I Mann
  • Jan 1957
  • 357-364
  • I Mann
Mann I. Development Abnormalities of the Eye. Lippincott: Philadelphia, 1957, pp 357-364.