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PubMed1979

Classification of oblique facial clefts with microphthalmia.

We believe the Tessier classification system facilitates the description of complicated facial clefts, and we describe its use in 4 cases--all of whom had variations of a Tessier-3 cleft. Microphthalmia is associated with the Tessier-3 cleft. Computerized axial tomography may be helpful in differentiating an extreme microphthalmia from a true anophthalmia, and in detecting bony clefts not seen on roentgenographic studies. Facial tomograms show that abnormalities of the medial wall of the orbit in the Tessier-3 cleft vary from actual clefts to widening of the ethmoid. In our experience (with 4 patients) mental retardation does not necessarily occur in patients with the Tessier-3 cleft and extreme microphthalmia. Abnormalities of the nasolacrimal apparatus may be the most constant finding in patients with the Tessier-3 cleft. Children with the Tessier-3 cleft should be identified early for purposes of determining the proper treatment and valid genetic counseling.

Anophthalmos↗
PubMed1987

Correction of enophthalmos and superior sulcus depression in the anophthalmic orbit: a long-term follow-up.

The purpose of this study was to review the long-term results in anophthalmic patients treated for enophthalmos. The patients in our series had augmentation of the volume of their orbital contents by subperiosteal placement of room-temperature vulcanized silicone. From 1972 to 1985, sixty-three patients received subperiosteal placement of room-temperature vulcanized silicone for their enophthalmos. Twenty-four of these patients were seen in long-term follow-up 1 to 13 years postoperatively. Indepth follow-up evaluation showed that room-temperature vulcanized silicone subperiosteal implantation for the treatment of enophthalmos and superior sulcus depression in the anophthalmic orbit is a reliable, safe procedure that is without serious complications and has excellent long-term results.

Adult↗
PubMed1996

The superior auricular artery and retroauricular arterial island flaps.

This paper reports the finding of the superior auricular artery, which is a fairly large arterial branch, about 1 mm in diameter, arising from the superficial temporal artery anterior to the auricular helix. It is covered by skin, the anterior auricular muscle, and the fascia of the parotid gland. It runs together with its concomitant vein posteriorly in the groove between the cartilage of the ear and the temporal bone and anastomoses with the posterior auricular artery. This artery has been used successfully to raise large retroauricular arterial island flaps for one case of total nose reconstruction and seven cases of reconstruction of the severely constricted anophthalmic socket. These operative procedures are described in detail.

Anophthalmos↗
PubMed1998

Eye socket reconstruction with the prefabricated temporal island flap.

Insufficiency of tissues and progressive contraction usually restrict the application of prosthetic devices in anophthalmic eye sockets. To achieve a successful reconstruction, the plastic surgeon has to form a socket that has proper dimensions and is completely covered by a well vascularized epithelial surface. Eye socket reconstruction with free skin, mucous membrane, or dermis-fat grafts usually remains unsatisfactory in severe cases. We have used a prefabricated temporal island flap to solve this difficult problem since 1983. In this method, a full-thickness skin graft is applied over the temporal fascia to create a prefabricated island flap based on the superficial temporal vessels. This flap is transposed into the eye socket 3 weeks later. Some modifications in flap design have been done to get better fitting of the prosthesis since that time. Thirty-three patients with constricted eye sockets that could not use prosthetic devices were treated with prefabricated temporal island flaps since 1983. The follow-up period was between 1 and 13 years. Eye sockets with adequate size and volume were created in all patients, and the results were successful. This method prevented secondary graft shrinkage, and the prefabricated island flaps preserved their dome shape during the follow-up period. We believe this method is a useful one in the treatment of the contracted socket.

Adolescent↗
PubMed1999

The direct effect of intraorbital pressure on orbital growth in the anophthalmic piglet.

Tissue expanders placed within the orbit can have a positive effect on orbital and ipsilateral midfacial growth. To date, there is no precise method for controlling and monitoring expansion to induce normal growth in the developing facial skeleton. The present study was undertaken to determine the optimal physiologic pressure required to stimulate normal orbital growth and to determine whether above-normal growth could be achieved with higher intraorbital pressures. Using a neonatal swine model, an accurate method of monitoring intraorbital pressure, precisely controlling intraorbital expansion, and achieving normal orbital growth was explored. Sixteen male, 3-week-old Yorkshire piglets were randomly divided into three surgical groups. In each group, the left orbit was the experimental side, and the contralateral right orbit served as an untreated control. Group 1 (n = 6) underwent enucleation only. Group 2 (n = 5) underwent enucleation and orbital expansion at a near-normal physiologic pressure of 20 mmHg. Group 3 (n = 5) underwent enucleation and orbital expansion at a supernormal pressure of 60 mmHg. Spherical tissue expanders (10 cc) with a separate injection port were utilized as the orbital expanders. Pressure was monitored by an electronic manometer that was calibrated daily. Morphology of the orbits was documented by photography, the dimensions of the orbits were quantitated by three-dimensional mechanical digitization, and orbital volumes were calculated. In the unexpanded, anophthalmic control group, a significant reduction in radial growth after evisceration was seen. In group 2, the orbit stimulated with a consistent pressure of 20 mmHg, just above the physiologic normal pressure of 17 mmHg, showed an increase in radial dimension of 8 percent compared with the unoperated side. In the high-pressure group of 60 mmHg, an increase of 16 percent in the radius was observed over the 4-week period. This led to a corresponding increase in orbital volumes with increased pressure. Utilizing a paired t test, these differences in the radial and volumetric growth of the orbit were statistically significant (p < 0.005). The results obtained demonstrated a direct relationship between intraorbital pressure and the growth of the bony orbit in the radial dimension. On the basis of this study, we concluded that orbital expansion maintained at normal physiologic pressure can stimulate normal orbital growth in the neonatal facial skeleton. In addition, application of above-normal pressures for expansion can induce accelerated orbital growth.

Animals↗
PubMed1980

Bilateral clinical anophthalmia: drugs as potential factors.

This case of bilateral clinical anophthalmia presents a host of interrelated drug reactions and potential teratogenic factors. While none of the drugs ingested by the mother has a high teratogenic potential, drug interactions, toxic potentiation, and unknown incompatibilities cannot be excluded. Although a single etiologic agent is not incriminated, the drugs ingested during pregnancy and their toxic effects and interactions are discussed. Use of drugs during pregnancy should be limited to those clinically indicated.

Abnormalities, Drug-Induced↗
PubMed1997

Oculoauriculofrontonasal spectrum in an adult Brazilian male.

We report an adult male patient born to normal and non-consanguineous parents with midline craniofacial defects associated with branchial arch anomalies. This combination of signs belongs to the oculoauriculofrontonasal spectrum. We compare our case with those previously reported who had similar findings and discuss clinical aspects of the oculoauriculofrontonasal spectrum and frontofacionasal dysplasia.

Adult↗
PubMed1997

Focal dermal hypoplasia (Goltz syndrome) presenting as a severe fetal malformation syndrome.

A fetal malformation syndrome comprising growth retardation, anophthalmia, bilateral diaphragmatic herniae, bifid lower leg, syndactyly of the fingers, malrotation of the colon, hypoplastic kidneys and total anomalous pulmonary venous drainage is described in a female fetus from a consanguineous relationship. Differential diagnosis is discussed and it is suggested that this case represents an unusually severe form of Goltz syndrome.

Abnormalities, Multiple↗
PubMed1998

Cerebro-oculo-nasal syndrome: another case and review of the literature.

Cerebro-Oculo-Nasal syndrome; a new multiple congenital anomaly/mental retardation syndrome was first reported by Richieri-Costa and Guion-Almeida in 1993 (Am J Med Genet 47:702-706) in two patients. To the best of our knowledge four additional cases have been reported. The main features of the syndrome are anophthalmia/microphthalmia, abnormal nares, and central nervous system anomalies. In this report, an additional sporadic case of this syndrome is presented. A 6-year-old girl from a non-consanguineous couple with normal prenatal growth parameters and retarded postnatal growth had anophthalmia, uplifted right nares with skin tag, and slight clefting at the tip of the nose, upper lip and gingiva. She also had a high-arched narrow palate, slightly low set ears, hypertelorism, a CNS defect and mental retardation. Additional findings were hypoplastic teeth with dental malocclusion, muscular hypotonia and midline hyperpigmentation over the anterior neck and the abdomen.

Abnormalities, Multiple↗
PubMed2003

Fryns "Anophthalmia-Plus" syndrome associated with developmental regression.

We report a boy with a nasal deformity, choanal atresia, bifid uvula, severe bilateral microphthalmia and a facial cleft who showed regression of development at the age of 2 years with subsequent improvement. We suggest he represents a further case of the rare Fryns "Anophthalmia-Plus" syndrome.

Abnormalities, Multiple↗
PubMed1993

Enucleation, evisceration, and exenteration.

This review outlines many of the recent advancements in the understanding and management of the anophthalmic patient. A population-based study demonstrated that the annual incidence of enucleations for all causes was about 3 to 5 per 100,000. Application of expandable orbital implants appears to be promising in the management of microphthalmia or anophthalmia in infants to maximize orbital growth. Some reports on the use of hydroxyapatite enucleation implants are encouraging, with no major complications observed in one large series. Yet other reports of hydroxyapatite implant exposures, at a very concerning frequency, are also beginning to emerge. Few of the exposures heal spontaneously; however, infections or extrusions are very rare, and they are attributable to the porous composition of the implant. Conjunctival flaps alone are suboptimal in the management of exposures. Adjunctive autologous fascial grafts seem preferable to heterologous sclera in the management of these exposures. Magnetic resonance imaging of the hydroxyapatite implant appears to be superior to bone scan in the noninvasive assessment of vascularization of these implants. Further advancements are necessary to achieve a more optimal enucleation implant.

Anophthalmos↗
PubMed2004

Management of the anophthalmic socket in pediatric patients.

PURPOSE OF REVIEW: The management of the pediatric anophthalmic socket is distinguished from adult anophthalmia, because normal socket and facial development is dependent on orbital growth. Recent literature on managing anophthalmic sockets is discussed with a focus on the pediatric patient. RECENT FINDINGS: There are many studies discussing the management of the anophthalmic socket, but few are randomized clinical studies. Although the management of anophthalmia in the child is fraught with challenges and nuances, the principal aim is to encourage socket growth. SUMMARY: Both congenital and acquired pediatric anophthalmia are discussed. Surgical decisions, choice of orbital implants, various methods of socket expansion, and socket reconstructions are examined.

Anophthalmos↗
PubMed2003

Anophthalmic ptosis: investigation of the mechanisms and statistical analysis.

PURPOSE: To investigate the mechanisms producing ptosis in anophthalmic patients by comparing potential risk factors among patients with ptosis with a control group. Specific techniques for achieving optimal aesthetic results in the surgical correction of anophthalmic ptosis are recommended. METHODS: Data from 94 anophthalmic patients with ptosis and 44 control patients without ptosis were retrospectively collected and analyzed. Potential risk factors for ptosis were compared between the two groups of patients by using 2-sample binomial exact tests. Univariate and multivariate logistic regression analyses were also used. RESULTS: A direct relation between percent volume replacement by the primary implant and the occurrence of ptosis was noted (P=0.057; age adjusted, P=0.037). Those patients who had secondary implant surgery were more likely to have levator surgery for ptosis (P=0.005; age adjusted, P=0.039). A greater number of years of prosthesis wear was associated with clinical and intraoperative evidence of levator dehiscence (P=0.061). No relation was observed between the occurrence of trauma (as a reason for enucleation) and the occurrence of levator dehiscence. Thirty percent of patients with ptosis who had secondary intraconal or extraconal implants and no other surgical intervention for ptosis had improvement in ptosis. CONCLUSIONS: The mechanisms producing anophthalmic ptosis should be assessed carefully before surgical repair to achieve optimal aesthetic results. Correction of volume deficiency should be offered before levator surgery for patients with anophthalmic ptosis. The patient should be advised that tilt of the prosthesis is a possible outcome of levator repair and may reflect overcorrection or insufficient volume replacement.

Adult↗
PubMed2004

Current trends in managing the anophthalmic socket after primary enucleation and evisceration.

PURPOSE: To evaluate current trends in the management of the anophthalmic socket after primary enucleation and evisceration. METHODS: The active membership of the American Society of Ophthalmic Plastic and Reconstructive Surgery (ASOPRS) was surveyed regarding primary enucleations and eviscerations performed between January and December 2002. Survey questions included practice demographics, orbital implant use, wrapping materials, placement of a motility peg, reasons for implant choice, and complications encountered. RESULTS: A total of 2,779 primary orbital implants were reported, comprising 1,919 (69.1%) enucleations and 860 (30.9%) eviscerations. The high-density porous polyethylene implant was used most frequently for enucleations (42.7%), followed by coralline hydroxyapatite (27.3%) and nonporous alloplastic implants (19.9%). For eviscerations, the high-density porous polyethylene implant was the most commonly used implant (42.3%), followed by hydroxyapatite (25.9%) and nonporous alloplastic implants (25.7%). The top 3 reasons for implant choice were outcome (69.3%), cost (43.6%), and experience (39.5%). Most implants were either not wrapped (59.8%) or were wrapped in donor sclera (25.2%) or polyglactin mesh (7.2%). Pegs were used in 8.1% of all implants reported. The most frequent complications encountered for unpegged implants were exposure (3.2%) and infection (0.4%). For pegged implants, the most common complications reported were pyogenic granuloma (13.7%), exposure (5.7%), and discharge (5.7%). CONCLUSIONS: In managing the anophthalmic socket, ASOPRS survey respondents preferred to use the porous polyethylene implant after primary enucleation and evisceration. Most orbital implants were not wrapped, and most surgeons preferred not to place a motility post or peg in the implant.

Anophthalmos↗
PubMed2004

Clinicopathologic analysis of 15 explanted hydroxyapatite implants.

PURPOSE: To report the clinical findings, treatment, outcomes, and histopathologic findings in patients with suspected orbital implant infection requiring implant removal. METHODS: Retrospective, observational case series of 14 patients (15 hydroxyapatite orbital implants) undergoing implant removal from September 1994 through December 2002. Patient age, type of surgery, implant type, symptoms, treatment, histopathology of implant, and follow-up course were analyzed. RESULTS: Of the 14 patients, 7 were female and 7 were male. The mean age at explantation was 42 years. The most common symptoms were discharge and socket tenderness. The most common signs were conjunctival inflammation (edema, hyperemia), discharge, and recurrent pyogenic granuloma. Clinical evidence of infection was documented in 13 patients. Histopathologic assessment of the 15 explanted implants showed acute inflammation and necrosis (abscess) with identification of microorganisms (5 patients), acute inflammation and necrosis without identification of microorganisms (4 patients), chronic inflammation with identification of microorganisms (1 patient), chronic inflammation without identification of microorganisms (3 patients), and a predominant foreign body granulomatous response without identification of microorganisms (2 patients). Osseous metaplasia was seen in 10 implants. Prompt resolution of symptoms and signs occurred in all but one case. CONCLUSIONS: The clinical course of porous orbital implant infection may be prolonged, and the early symptom of recurrent discharge, a common problem for implant recipients, may delay diagnosis. Implant infection should be suspected when there is persistent conjunctival inflammation and discharge after implant placement despite antibiotic therapy, discomfort on implant palpation, and recurrent pyogenic granuloma (indicative of implant exposure). Implant removal is usually required in these cases. If orbital pain (not necessarily related to implant palpation) is the main complaint, without signs of conjunctival inflammation and with or without discharge, one should consider other reasons for the symptoms.

Adolescent↗