EditorialLa retina como marcador biológico de daño neuronalArtículos originalesComparación de tres instrumentos de tomografía de

coherencia óptica, un time-domain y dos Fourier-domain, en la estimación del grosor de la capa de fibras nerviosas de la retina

Idoneidad de tratamiento en sospechosos de glauco-ma. Estudio de concordancia con el grupo de estu-dio RAND

Atrofia de la capa de fibras nerviosas de la retina en pacientes con esclerosis múltiple. Estudio prospec-tivo con dos años de seguimiento

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Short communication

Neurotrophic corneal ulcer in an HIV patient☆

J. Paz Moreno-Arrones,a,b,* J. Benítez-Herreros,a,b P. Drake-Rodríguez,a,b A. Romero-García Tenoriob,c

aUniversidad de Alcalá de Henares, Alcalá de Henares, Madrid, Spain bHospital Universitario Príncipe de Asturias, Madrid, Spain cUniversidad Complutense de Madrid, Madrid, Spain

ARTICLE INFORMATION

Article history:

Received on Aug. 2, 2010

Accepted on Oct. 26, 2010

Keywords:

Tarsorrhaphy

Neurotrophic ulcer

Cerebral toxoplasmosis

☆Presented at the 85th Congress of the Spanish Ophthalmological Society. *Corresponding author.

E-mail: javier_paz_moreno@hotmail.com (J. Paz Moreno-Arrones).

A B S T R A C T

Case report: We present the case of a 29 year-old man who came to the Emergency

Department due to pain in the right eye. There was demonstrated a complete corneal de-

epithelialisation. There was no clinical improvement after appropriate treatment, which

was complicated by migraine and vomiting. The computerized tomography (CT) scan

showed images suggestive of cerebral toxoplasmosis. After the complete tarsorrhaphy a

restitution ad integrum was observed.

Conclusion: The diagnosis of a neurotrophic corneal ulcer due to an affected trigeminal

nerve in the context of a cerebral toxoplasmosis, tarsorrhaphy is an effective procedure to

take in account in corneal epithelial defects resistant to other treatments.

© 2010 Sociedad Española de Oftalmología. Published by Elsevier España, S.L.

All rights reserved.

Úlcera corneal neurotrófica en paciente con VIH

R E S U M E N

Caso clínico: Se presenta el caso de un varón 29 años acude a Urgencias de oftalmología por

dolor en ojo derecho. Se evidenció una desepitelizacion corneal completa. Tras un trata-

miento adecuado no se evidenció mejoría clínica, complicándose el cuadro con cefalea y

vómitos. la tomografía axial computarizada (TAC) evidenció imágenes sugestivas de toxo-

plasmosis cerebral. Tras la tarsorrafia completa se observó una restitución ad integrum del

cuadro.

Conclusión: Ante el diagnóstico de una úlcera corneal neurotrófica por afectación del trigé-

mino en el contexto de toxoplasmosis cerebral, la tarsorrafia es un procedimiento eficaz a

tener en cuenta ante defectos epiteliales corneales resistentes a otros tratamientos.

© 2010 Sociedad Española de Oftalmología. Publicado por Elsevier España, S.L.

Todos los derechos reservados.

Palabras clave:

Tarsorrafia

Úlcera neurotrófica

Toxoplasmosis cerebral

28 ARCH SOC ESP OFTALMOL. 2011;86(1):27–30

Introduction

Neurotrophic ulcers consist of a degenerative corneal condition caused by any noxa that affects corneal sensitivity. The absence of corneal sensitive trophic effects entails the impossibility of corneal healing which associates an epithelial defect, ulceration and even perforation.

Tarsorrhaphy is a simple, efficient and safe procedure for managing various ocular surface conditions such as neurotrophic ulcers due to the involvement of the trigeminal nerve, facial paralysis, lagophthalmos among others, carried out in isolation or in combination with other ocular plastic surgical techniques.

Clinical case

African male, 29, who visits the ophthalmological emergency section due to pain and diminished vision in the right eye (RE). The exploration revealed a visual acuity (VA) of finger counting at 3 m in RE which did not improve with the stenopeic hole, and of 1 in the left eye (LE).

In the anterior biomicroscopy a ciliary injection was observed with complete corneal de-epithelization, stromal edema and folds in Descemet membrane (fig. 1), with the rest of the ophthalmological exploration being normal. It was decided to initiate an occlusive treatment with cycloplegic eyedrops comprising antibiotics. Two days later, apart from not evidencing improvement in the ocular condition, the patient associated holocranial headache and vomiting. It was decided to maintain the ophthalmological treatment and to refer the patient to the internal medicine emergencies for a full assessment. After a systemic exploration, diminished facial sensitivity was evidenced in the fourth branch of the trigeminal, with corneal anesthesia. A computerized axial tomography (CAT) was taken which evidenced triventricular hydrocephalia, with two hypodense areas being observed in

the right cerebellum hemisphere and temporal lobe (fig. 2), compatible with edema areas. Internal medicine established antitoxoplasmic oral treatment together with ganciclovir. The magnetic resonance imaging (RMI) with contrast evidenced several space-occupying lesions in the right and left cerebellum hemispheres, in the temporal lobe and the frontal operculum of the left islet surrounded by edema (fig. 3) suggesting cerebral toxoplasmosis. The lumbar puncture yielded normal results. In an additional anamnesis, the patient referred he was HIV-positive for 2 years due to a high risk sexual relationship. He did not refer consuming

Figure 1 – De-epithelization with stromal edema and folds in Descemet membrane.

Figure 2 – The CAT images shows a general blurriness of the cerebellum grooves with hypodense areas compatible with areas of edema.

Figure 3 – RMI image showing lesions with different sizes surrounded by vasogenic edema. The ring-shaped capture of lesions is marked with arrows.

ARCH SOC ESP OFTALMOL. 2011;86(1):27–30 29

Figure 4 – Image showing the partial tarsorrhaphy of the 2 external thirds of the eyelid and the improved appearance of the ocular surface.

drugs. In the light of this finding, the following serological tests were carried out: ELISA and Western blot for HIV, IgM and IgG for toxoplasmosis, with positive results. The diagnostic was cerebral toxoplasmosis in patient with HIV. The lymphocyte T CD 4+ was of 69/μl, with a number of HIV-RNA copies of 147.460/ml. Our ophthalmological diagnostic was neurotrophic ulcer due to involvement of the trigeminal in the context of cerebral toxoplasmosis. As no ophthalmological improvement was evidenced with the previous treatment, autogenous serum and therapeutic contact lens was prescribed without achieving the resolution of the ophthalmological clinical condition. Due to the slow evolution of the corneal ulcer, paralytic ptosis was induced by injecting 12 international units of botulin toxin at the level of the RE upper eyelid elevator. However, due to the poor cooperation of the patient, who continually raised the eyelid in a reflex action, the ulcer failed to heal. Finally it was decided to carry out a permanent tarsorrhaphy of the two external thirds in the RE, suturing the anterior and posterior lamellae of both eyelids and treating with autogenous serum, antibiotic eye drops and cycloplegic, upon which the patient evidenced clinical improvement (fig. 4).

Discussion

The cornea is a highly sensitive structure that is able to perceive different feelings such as pain, heat, cold or touch. The absence of corneal sensitivity involves a disruption of the integrity of the corneal surface.

A neurotrophic keratoplasty is a clinical entity which comprises all the degrees of corneal and conjunctival degeneration secondary to the loss of the sensitive function in the pathway of the nasocilliar branch (V1) of the trigeminal nerve with or without diminished tear production.

The most frequent etiology of the clinical condition known as corneal anesthesia is the infection of the corneal surface by Herpes Simplex and Herpes Zoster.1 It is known that corneal sensitive nerves play a crucial role in maintaining the anatomic and functional integrity of the cornea and its epithelium, even though the exact mechanism is yet not fully determined. It is known that the damage of these nerves causes a reduction in metabolism and mitosis of epithelial cells together with an increase of their permeability.2,3 The corneal epithelium proliferation is probably regulated by a two-way control, i.e., sensitive neuromediators that promote mitosis in epithelial cells and sympathetic mediators that reduce them. As a result of this neurologic disruption, in neurotrophic ulcers the epithelium defect persists and therefore cannot heal. However, if the ocular surface is protected from the environment with therapeutic contact lenses or tarsorrhaphy, the ulcer almost always heals.

It must be noted that heroine addicts can evidence false corneal neurotrophic ulcers either to direct inoculation of heroine in their eyes as a result of manipulating or inhaling it, which can be difficult to diagnose.

The neurological complications of the human immunodeficiency syndrome (AIDS) are frequently symptoms of opportunistic infections of the central nervous system (CNS), as in this case toxoplasmosis. The reactivation of the latent infection occurs in patients having their immune system compromised, mainly causing meningoencephalitis,4 but it can also cause a condition of polyradiculoneuritis and miositis. Similarly, cerebral toxoplasmosis is the most frequent cause of space-occupying lesions in AIDS patients (typically in advanced stages or in patients with less than 200 lymphocytes T CD4+/μl) characterized by being lesions with mass effect, affecting the basal ganglions. The definitive diagnosis requires brain biopsy which, due to its high morbidity, is reserved only for cases that do not improve with empirical treatment. This clinical condition is more frequent with lower lymphocyte T CD 4+ counts. In clinical practice, the diagnostic and treatment for cerebral toxoplasmosis are initially presumptive, based on clinic and radiological findings.5

The determination of IgM and IgG positive antibodies at the plasma level established the definitive diagnostic of this disease even when this serology is negative at the level of the cerebrospinal fluid because up to one third of cerebral toxoplasmosis patients exhibit repeated negative IgG in the lumbar puncture. However, it must be noted that with these patients we must take into account the differential diagnosis with other diseases affecting the central nervous system such as progressive multifocal leucoencephalopathy, criptococcic meningoencephalitis and malign cerebral lymphoma.

The «HAART» antiretroviral therapy has demonstrated improvements in the efficacy of anti-toxoplasma drugs through our recovery of the immune system, in addition to reducing mortality in HIV-positive patients.

In cases involving neurotrophic corneal ulcers the treatment of the epithelial defects must be established as soon as possible in order to prevent its progression to corneal perforation and the sequels this produces. It must be remembered that, in these cases of severe corneal anesthesia, penetrating keratoplasty exhibits poor results, even after a

30 ARCH SOC ESP OFTALMOL. 2011;86(1):27–30

corneal transplant. Thus, the therapeutic alternatives consist in occlusion with patches, placement of contact lenses, botulin toxin injection in the upper eyelid elevator, translocation of a conjunctival flap to the cornea, amniotic membrane transplants and, in the failure of these techniques, the last option to be considered is either temporary or permanent tarsorrhaphy.

In our case, due to the brief period of action exhibited in this patient at the corneal level surface, the amniotic membrane transplant was not utilized. However, as a result of the torpid evolution of corneal de-epithelization which resisted different conservative treatments, it was decided to form a permanent tarsorrhaphy of the 2 external thirds, previously separating the anterior and posterior lamellae of both eyelids to join them together, adding cycloplegic and antibiotic eyedrops and achieving adequate corneal epithelization.

We would like to emphasize the need of establishing this type of treatment if conservative therapeutic efforts fail in these conditions.

Conflict of interest

None of the authors have declared any conflict of interest.

R e f e r e n c e s

1. Groos EB. Neurotrophic tis. In: Krachmer JH, Mannis MJ, Hooland EJ, editors. Cornea: Fundamentals of corneal and external disease. St. Louis: Mosby; 1997. p. 1339-62.

2. Sigelman S, Friedenwald JS. Mitotic and wound healing activities of the corneal epithelium: effect of sensory denervation. Arch Ophthalmol. 1954;52:46-57.

3. Simone S. De ricerche sul contenuto in acqua totale ed in azoto totale della cornea di coniglio in condizioni di cheratite neuroparalitica sperimentale. Arch Ottalmol. 1958;62:151.

4. Hill D, Dubey JP. Toxoplasma gondii: transmission, diagnosis and prevention. Clin Microbiol Infect. 2002;8:634-40.

5. Cohen B. Neurological manifestations of toxoplasmosis in AIDS. Semin Neurol. 1999;19:201-11.