CASE REPORT |
https://doi.org/10.5005/jp-journals-10077-3297 |
Overhauling the Anophthalmos due to Retinoblastoma in Pediatric Patient: A Case Report
1,3Department of Paediatrics and Preventive Dentistry, Government College of Dentistry, Indore, Madhya Pradesh, India
2,4Department of Prosthodontics, Crown & Bridge and Implantology, Government College of Dentistry, Indore, Madhya Pradesh, India
Corresponding Author: Prajakta Anadeo, Department of Prosthodontics, Crown & Bridge and Implantology, Government College of Dentistry, Indore, Madhya Pradesh, India, Phone: +91 8319213711, e-mail: prajaktaanadeo123@gmail.com
Received: 28 February 2024; Accepted: 21 March 2024; Published on: 27 April 2024
Anophthalmos, which means ”absence of eye,” is not only a physical or physiological but also a psychosocial burden on a growing child. Retinoblastoma has been found to be the most common tumor of ocular origin, management of which often includes enucleation of the affected eye, rendering the patient disabled. This article is intended to emphasize the rehabilitation of the enucleated left eye due to retinoblastoma by presenting a case of a 5-year-old child restored with a noninvasive customized ocular prosthesis fabricated by modifying the stock eye to achieve a comfortable fit, slight motility, and esthetics. The patient was assessed for compliance, level of satisfaction, and the need for replacement of the prosthesis at follow-up periods of 1, 3, and 6 months. This article encourages pediatric dentists to successfully manage minor patients and prosthetically rehabilitate the orofacial defects to facilitate the development of the orbital region and enhance the self-esteem of the child. How to cite this article: Anadeo P, Gupta A, Saxena A, et al. Overhauling the Anophthalmos due to Retinoblastoma in Pediatric Patient: A Case Report. J South Asian Assoc Pediatr Dent 2024;7(1):46–49. Source of support: Nil Conflict of interest: None Patient consent statement: The author(s) have obtained written informed consent from the patient’s parents/legal guardians for publication of the case report details and related images.ABSTRACT
Keywords: Anophthalmos, Case report, Enucleation, Ocular prosthesis, Retinoblastoma, Self-Esteem
INTRODUCTION
The eyes are the very first organs to be noticed in an individual. The absence of an eyeball has a great impact on a child’s self-esteem and severely impedes the growth and development of the facial region. The various causes of a missing eyeball may be congenital, including agenesis, deformities or acquired, including malignancy, trauma, infection, etc.1 Malignancy, especially retinoblastoma, is the most common cause where removal of the eyeball is indicated in pediatric patients.2 The treatment mainly includes evisceration, enucleation, and exenteration depending upon severity or invasion, followed by rehabilitation with fixed (ocular implants) or removable (stock, modified stock, custom-made) ocular prostheses. The purpose of an ocular prosthesis is to preserve the anophthalmic space, partially reestablish the lachrymal position, prevent fluid accumulation, restore facial esthetics, promote the growth of the orbit, and boost the patient’s self-esteem. As the child has been exposed to the surgery earlier, he/she is usually fearful of the hospital set-up, making the fabrication of the ocular prosthesis a challenging process. The prosthesis is recommended to be replaced every 18–24 months as the child grows in order to effectively stimulate the development of surrounding tissues and minimize abnormalities.3 This validates the role of a pediatric dentist who understands a child’s psychology and growth very precisely.
According to a study conducted by Dahan), the instances of missing eyeballs in pediatric patients in the decreasing order of occurrence were as follows—retinoblastoma, trauma, microphthalmia, anophthalmia, endophthalmitis, congenital glaucoma, etc.4 Retinoblastoma is highly prevalent in Asian countries, with India having the highest anticipated incidence of retinoblastoma in the world, with about 1,400 out of 8,000 new cases diagnosed annually in children below 5 years of age. In order to rehabilitate the enucleated eyeball to maintain the tissue integrity as well as promote orbital growth, it is important for a pediatric dentist to have a thorough knowledge of the selection and fabrication of the ocular prosthesis.
The stock eye prostheses which were used earlier are simple, easy, and less time-consuming, but the discomfort and potential for bacterial infections due to water pockets and incorrect fit make them obsolete and pave the way for customized ocular prosthesis, which require longer laboratory time for its fabrication. Thus, the modified stock eye technique by Taicher et al.5 in 1985 was a simple yet effective alternative to the uncomfortable stock and time-consuming custom ocular prostheses. This article reports a case of prosthetic rehabilitation with a modified stock eye for a 5-year-old patient with an enucleated left eyeball due to retinoblastoma.
MATERIALS AND METHODS
A 5-year-old patient presented in the outpatient Department of Pediatric and Preventive Dentistry with a chief complaint of unesthetic facial appearance due to a missing left eye (Fig. 1). The patient lost her eye due to enucleation of the eyeball invaded by retinoblastoma. After a thorough clinical examination of the eye socket and explaining all the possible treatment modalities, including removable stock, custom eye prosthesis, implant-supported prosthesis, retinal prosthesis, etc., to the patient’s mother, it was decided to plan prosthetic rehabilitation of the missing eye with modified stock eye.
Figs 1A and B: Preoperative photograph; (A) Front view; (B) Enucleated satisfactorily healed socket
The patient was made comfortable with verbal and nonverbal behavior management techniques, and the child was made to sit upright in a physiologic rest position. The scleral shell (stock eye) was selected according to the patient’s iris color and size so that it could be used in the final prosthesis. It was further modified to comfortably fit the left eye socket. The modified stock eye was duplicated with autopolymerizing polymethyl methacrylate (DPI-RR; Wallace Street, Mumbai, Maharashtra, India) to fabricate the acrylic tray (Fig. 2A). In order to prevent soft tissue irritation, the extension of this resin tray was evaluated in the patient’s eye socket. The disposable syringe needle cap was attached to the fabricated resin tray at a position of the pupil to use it as a handle and flow the material. Escape holes were created to aid in the outflow of the excess impression material. Petroleum jelly was coated along the socket and periorbital tissues, including eyelid, eyelashes, and eyebrows, to promote easier retrieval of the impression material.6 Light body rubber-based impression material (Aquasil Ultra XLV; Dentsply DeTrey, Konstanz, Germany) was loaded on the tray and was gradually infused into the socket to avoid air voids as well. The patient was instructed to sit erect, have a straight gaze and perform all the functional eye movements to allow the material to flow into all the extensions.7
Figs 2A to D: Fabrication of the prosthesis; (A) Duplication of the scleral shell followed by attaching the handle to the duplicated acrylic tray; (b) Recorded impression; (C) Pouring of the cast with two pour technique; (D) Positioning of the iris on the poured wax pattern (I, inferior border; L, lateral; M, medial; S, superior)
The impression was gently retrieved, as shown in Figure 2B, and poured using the two-pour technique. The tissue surface was poured with a die stone (type IV gypsum product), and the second pour was made with dental stone (type III gypsum product). A separating medium was applied between the two pours; index markings were made before the first pour was partially set to guide in further steps, as shown in Figure 2C.8 The disposable syringe needle cap was retained to act as a sprue for pouring the molten wax. The molten wax was poured onto the cast through this sprue, and the iris taken out from the stock eye was positioned on the wax pattern (Fig. 2D).
The reference landmarks to best resemble the contralateral eye were marked as shown in Figure 3. The wax pattern was tried in the patient’s eye socket, and the position of the iris, scleral contour, prosthesis extension, palpebral movements, palpebral fissure opening, etc., was assessed.
Fig. 3: Iris positioning with all the reference marking
The adjusted wax pattern was invested in a specially designed flask, and dewaxing was carried out by securing the iris with the help of a toothpick to one compartment of the flask. Heat cure acrylic resin (Stellon, Dental Products of India Ltd.) of the matched shade was employed, and stains and flocks were added to give a more natural appearance before final closure. The prosthesis was then processed with a slow curing cycle after the final closure. The recovered prosthesis was finished and polished for the final insertion. The prosthesis was inserted, adjusted if required, and assessed for esthetics and patient comfort (Fig. 4).9 The patient was educated and motivated using audiovisual aids to clean, insert and remove the prosthesis. The patient’s parents were instructed to insert the prosthesis by lifting the upper eyelid while removing the prosthesis by pulling the lower eyelid and maintaining prosthesis hygiene.10 The patient was emphasized on regular recall visits and was informed of the possible need to change the prosthesis in 18–24 months.3 Lubricant (0.5% sodium carboxymethylcellulose) eyedrops, one drop at bedtime, were advised to the patient.
Fig. 4: Removable ocular prosthesis insertion
DISCUSSION
Retinoblastoma has a high prevalence (2% of all pediatric cancers) in children and is frequently aggressive and invasive in nature.11 The treatment mainly includes evisceration, enucleation and exenteration. Evisceration includes the removal of internal contents of the eye, enucleation is the removal of the complete eyeball, and exenteration involves the removal of the eyeball along with the extraocular muscles.12,13 Enucleation was carried out in this patient as the tumor involved the left optic nerve along with the eyeball. This disability is prosthetically rehabilitated with either fixed ocular implants or removable ocular prostheses. A removable ocular prosthesis was selected for this patient due to technical and financial constraints. Removable ocular prosthesis can be either stock eye prosthesis or custom eye prosthesis. In order to overcome the demerits of stock (improper fit, discomfort, and bacterial accumulation due to voids) as well as the custom eye prosthesis (longer laboratory time), a simple yet effective modified stock eye prosthesis technique suggested by Taicher et al. was used for the fabrication of prosthesis in the presented case.5,14
Polymethyl methacrylate was the material of choice for the fabrication of an ocular prosthesis in this case due to its esthetics, adaptability, strength, longevity and possibility of repair and remodeling, which is required as the patient was in her growing phase.
The patient reported for rehabilitation after 9 months of enucleation with a shrunken eye socket. Rehabilitation with an ocular prosthesis is necessary as it has been proven to promote the growth and development of periorbital tissues. The marked esthetic correction of gross facial asymmetry built the confidence and boosted the child’s self-esteem, ultimately enhancing the child’s quality of life.
Various studies have been conducted to determine the growth of orbit according to which rapid growth occurs till 12 years of age. In order to keep pace with a child’s growth, orbital prosthesis requires regular replacements, which can be assessed by regular follow-ups. According to continuous growth leads to the loosening of the prosthesis and needs to be replaced every year. The patient will be assessed for the common signs of ocular prosthesis modification/replacement in juvenile patients (including improper fit, rotation of prosthesis inside the socket, malpositioned cornea, enophthalmic prosthesis, esthetically significant ptosis), patient comfort, satisfaction, and discoloration in the regular follow-ups.15
At 1 and 3-month follow-ups, there was adequate parent satisfaction, patient compliance and color stability were acceptable, while surface roughness was noticed at the 6-month follow-up. Long-term follow-ups could only be able to determine the color stability, patient compliance, and time for replacement of the prosthesis in pediatric patients, which is not available in the presented case report. The prosthetic ocular implants have better compliance and longevity than the removable ocular prosthesis. Also, the rehabilitation does not restore the physiology of the eye for which various novel modalities have been discovered like retinal prosthesis: bionic eye, three-dimensional artificial retina, EyePrintPro, etc.16
The child was fearful at the time of the ocular impression, and only after the incorporation of the behavior management techniques was she ready to cooperate and accept the treatment procedure. Pediatric dentists have a better understanding of child psychology and are capable enough to make the child cooperate during the clinical procedures of prosthesis fabrication like impression, wax try-in, iris positioning, etc., which are possibly scary for a child as they are to be done directly in front of the patient’s eyes. This simplified yet customized prosthesis can be easily fabricated by pediatric dentists and broadens the scope of pediatric dentistry in the realm of oral and maxillofacial prostheses for children.
CONCLUSION
An ocular prosthesis for pediatric patients provides an incredible psychological and social advantage as an adjunct to esthetics. Patient education and motivation aid for better cooperation of minor patients during the clinical procedures required in the fabrication of the prosthesis. The modified stock prosthesis technique was a time-efficient, esthetically sound, cost-effective, and highly acceptable technique for this patient. The patient is recalled every 3 months for follow-up and assessment for relining or replacement needs.
ORCID
Prajakta Anadeo https://orcid.org/0009-0006-8600-2833
Ashish Saxena https://orcid.org/0000-0001-9846-7095
ACKNOWLEDGMENT
I am thankful to the Department of Prosthodontics, Crown & Bridge and Implantology for the support throughout the process of fabrication of the prosthesis.
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