Orbital cellulitis and osteomyelitis secondary to odontogenic infection with campylobacter rectus: a case report.

The authors report the first case of orbital osteomyelitis due to Campylobacter in a 50-year-old male on a background of poor dental health. Campylobacter rectus is a member of the human oral flora and is usually associated with periodontal disease. There are 16 reported cases of non-oral C. rectus invasive soft-tissue infections, of which only one reports of osteolytic changes. In our patient, it is hypothesised that contiguous spread of periodontal infection with C. rectus seeded infection to the orbit. C. rectus infection is a rare but significant pathogen that should be considered as the etiologic factor in a patient presenting with an orbital lesion and bony changes, particularly on a background of poor dentition.

Clinical and Radiological Features of Intramuscular Orbital Amyloidosis: A Case Series and Literature Review.

PURPOSE: Orbital amyloidosis of the extraocular muscles (EOMs) is a rare condition, and its clinicoradiological features are not well elucidated. This study describes the characteristic clinical signs, MRI features, and potential treatment options. METHODS: Retrospective multicenter case series and literature review of EOM amyloidosis. RESULTS: Five cases were identified for inclusion. Common clinical findings were diplopia, ophthalmoplegia, and proptosis. Systemic amyloidosis was more likely to present with multiple muscle involvement, but no particular pattern was observed with localized disease. On MRI, amyloid deposition was characterized as a heterogeneous intramuscular mass with T2 hypointensity and post contrast enhancement. Management is dependent on the extent of disease and functional impairment; options include surgical debulking and radiation therapy. CONCLUSION: EOM amyloidosis is uncommon. The combination of clinical and radiologic findings described in this study should lead to its clinical suspicion.

A practical guide on the use of imiquimod cream to treat lentigo maligna.

Lentigo maligna (LM) is a common in situ melanoma subtype arising on chronically sun-damaged skin and mostly affects the head and neck region. Localisation in cosmetically sensitive areas, difficulty to obtain wide resection margins and advanced patient age/comorbidities have encouraged investigation of less invasive therapeutic strategies than surgery in managing complex cases of LM. Radiotherapy and imiquimod have emerged as alternative treatment options in this context. The treatment of LM with imiquimod cream can be challenging due to the nature of the disease including its often large size, variegated appearance, involvement of adnexal structures, poorly defined peripheral edge and frequent localisation close to sensitive structures such as the eyes and lips, and elderly patients with multiple comorbidities. Prolonged and unpredictable inflammatory reaction and side effects and compliance with a patient-delivered therapy can also be challenging. In the literature to date, studies evaluating the use of imiquimod to treat LM have utilised varying methodologies and provided short follow-up and these limitations have impaired the development of clear guidelines for dosage and management of side effects. Based on our multidisciplinary experience and review of the literature, we propose practical clinical strategies for the use of imiquimod for treating LM, detailing optimal administration procedures in various clinical scenarios and long-term management, with the aim of facilitating optimal patient outcomes.

Lower eyelid entropion repair with retractor mobilization and insertion onto the anterior surface of the tarsal plate.

PURPOSE: To evaluate a modified surgical technique for the correction of lower eyelid involutional entropion in terms of recurrence rate and relief of symptoms. METHOD: A prospective series of 67 consecutive eyelids in 55 patients who underwent lower eyelid entropion repair using a modified surgical approach not previously published in the literature to the authors' knowledge. The surgical technique, via a skin crease incision, involves disinsertion of the lower eyelid retractors from the tarsus and conjunctiva and suturing the retractors onto the anterior surface of the tarsal plate. Standard excision of redundant lower lid skin and orbicularis muscle was also performed as well as lateral canthal tendon repair. All procedures were performed by a single surgeon (B. A. O'donnell). This study adhered to the principles of the Declaration of Helsinki. RESULTS: There were 67 eyelids with a minimum of 12 months follow-up: of these only 1 patient (1%) required revision surgery (average follow-up 24 months). Four other eyelids had persistent or recurrent irritation (6%) not suggestive of, or secondary to, recurrence. Seven eyelids (10%) had undergone previous operative repair with no further recurrence following their most recent surgery. CONCLUSION: Disinsertion and reattachment of the retractors to the anterior tarsal plate in lower eyelid entropion repair is an effective surgical technique to improve lower eyelid stability. Although minimum follow-up is 12 months and average follow-up 24 months, longer follow-up would further evaluate this technique.

Non-Exenteration Management of Sino-Orbital Fungal Disease.

PURPOSE: To describe the non-exenteration management of sino-orbital fungal infection, a life-threatening condition for which orbital exenteration is generally considered a first-line treatment. METHODS: A retrospective case series is presented of 7 orbits in 6 consecutive patients admitted and treated at 2 major metropolitan tertiary teaching hospitals in Sydney, New South Wales, Australia. RESULTS: Seven orbits in 6 consecutive patients with sino-orbital fungal infection were treated conservatively with surgical debridement and intravenous antifungal agents. Four patients were immunosuppressed and the other 2 patients were otherwise healthy. All presented with pain, proptosis, or loss of vision. Causative organisms found were Mucormycoses, Aspergillus, and Scedosporium prolificans. Exenteration was avoided in all patients as part of their planned management and 5 patients, including 1 with bilateral disease, survived their disease without exenteration. Medical treatment included intravenous liposomal amphotericin B or voriconazole. A single immunosuppressed patient deteriorated and as a last resort, exenteration was performed, but this made no difference to his clinical course and in retrospect could have been avoided as he died of multiple cerebral metastases diagnosed shortly after his deterioration. CONCLUSION: The authors recommend that patients with sino-orbital fungal disease preferably be treated conservatively, without orbital exenteration.

Treatment and outcomes of malignant melanoma of the eyelid: a review of 29 cases in Australia.

PURPOSE: To review the treatment and outcomes of malignant melanoma (MM) of the eyelid skin. DESIGN: Retrospective case series review. PARTICIPANTS: All consecutive patients who had MM arising from eyelid skin treated by 2 regional tertiary referral oculoplastic surgeons were included. METHODS: Patient charts were reviewed to collect information on the main outcome measures. MAIN OUTCOME MEASURES: Demographics, clinical and histological features of the lesion, treatment, and outcomes. RESULTS: Twenty-nine patients between 22 and 88 years old (mean, 65) were included. The most common site of MM occurrence was the lower eyelid. Seventeen cases arose in an area of pigmentation, 4 arose de novo, and 8 were of unknown origin. The most common histopathological types were lentigo maligna melanoma (19 cases), followed by superficial spreading MM (8 cases). Fourteen patients had in situ disease and therefore had no Breslow thickness. Another 7 patients had Breslow thickness of <0.76 mm. Thirteen patients had Clark level II or higher. According to the American Joint Committee on Cancer staging system for cutaneous melanoma, 14 patients were clinically stage 0 and 6 patients were stage IA, with thickness < or = 1 mm and no ulceration. Treatment included wide excision in all cases, one of which underwent anterior exenteration. Pathological techniques used included mapped serial excision with standard or overnight paraffin sections or Mohs' micrographic surgery. Most patients had a good outcome, although 2 died of the disease. Five patients had local recurrence, and 4 had distant metastases. Median postoperative follow-up was 3 years (range, 1 month-9 years, 9 months). CONCLUSIONS: Lentigo maligna melanoma compared with other forms of MM was relatively more common in the periocular region than in other body locations. Our pathologists preferred paraffin sections to frozen section for accurate assessment of melanocytic atypia and margin status. Initial wide excision margins of 10 mm from the macroscopic edge of the tumor are suggested, as histological margins may be less than this. Margin control by mapped serial excision or a modified Mohs' micrographic surgery using paraffin sections is a useful technique to ensure complete excision and minimization of local recurrence.

Idiopathic sclerosing orbital inflammation.

OBJECTIVE: To perform a multicenter review of the clinical features and treatment of 31 patients with idiopathic sclerosing orbital inflammation. METHODS: We included all patients with histologically confirmed idiopathic sclerosing orbital inflammation from 5 regional orbital centers. We reviewed the case notes to determine the clinical presentation, diagnostic features, and response to treatment. The main outcome measures were duration and nature of symptoms, anatomical location of disease, histopathological findings, treatment modalities, treatment efficacy and adverse effects, and final clinical status. RESULTS: We included 13 male and 18 female patients ranging in age from 7 to 83 years. The average duration of symptoms at presentation was 13.4 months. There was a predilection for the lateral and superior quadrants. Thirteen patients had apical disease, and 4 had extraorbital involvement. Histopathological findings invariably showed sclerosis associated with a sparse mixed cellular infiltrate. Twenty-seven patients were treated with oral prednisolone, response to which was good in 9 patients, partial in 11, and poor in 7. Six patients were treated with a second-line immunosuppressive agent, and 6 received radiotherapy. The response to radiotherapy was generally poor. CONCLUSIONS: Idiopathic sclerosing orbital inflammation is a rare condition that can be difficult to diagnose and manage. Early intervention with immunosuppression in the form of corticosteroids combined with second-line agents can result in control and even regression of the disease.

Assessing patients with epiphora who are patent to syringing: clinical predictors of response to dacryocystorhinostomy.

PURPOSE: To assess the value of an abnormal fluorescein dye disappearance test (FDDT), lacrimal syringing, and Jones test for patients with epiphora who are clinically patent to syringing. METHODS: Prospective cohort study of 68 consecutive patients with epiphora who were clinically patent to syringing and otherwise normal to examination. Patients were assessed using FDDT and lacrimal syringing, as well as the Jones test comparing either single- or multiple-drop technique. FDDT and canalicular reflux on lacrimal syringing were subjectively graded. RESULTS: Success was defined as nil or only mild epiphora after surgery. Surgery was performed on 68 patients with a successful result in 64 (94%). The majority of these patients had severely delayed FDDT (90%), > or = 50% reflux on lacrimal syringing (78%), or were Jones I negative (81%). There was no significant difference between outcomes of the single- and multiple-drop tests. In patients examined with the single-drop Jones test, patients with a Jones I negative result had a statistically significant better surgical outcome (p = 0.04). This comparison was highly significant when the subgroup of patients with severely delayed/nonclearing FDDT and > or = 50% reflux was examined (p = 0.005). The results were not significant for the multiple-drop group. CONCLUSIONS: These findings show that a negative single-drop Jones I test is predictive of symptomatic improvement after dacryocystorhinostomy surgery in patients with epiphora who are clinically patent to syringing. Lacrimal syringing and the FDDT, on their own, were not predictive of surgical outcome. Jones testing was of significant value in patients, but only when the traditional single-drop test was used.

Medial canthal tendon release and lower eyelid retractor plication for Centurion syndrome.

Centurion syndrome is an uncommon, idiopathic medial canthal anomaly that causes epiphora due to the forward displacement of the lacrimal punctum out of the tear lake associated with the abnormal anterior insertion of the medial canthal tendon and enophthalmos. A case of Centurion syndrome is presented together with the description of a previously unreported surgical treatment: the combined surgical techniques of medial canthal tendon release and lower eyelid retractor plication.

Secondary tarsoconjunctival flap after previous lower eyelid Hughes repair.

PURPOSE: To describe a secondary, two-stage eyelid-sharing tarsoconjunctival flap advancement technique for the repair of full-thickness lower eyelid defect, using the remaining upper eyelid marginal tarsus that was spared in a previously performed Hughes procedure. METHODS: A computerized database search was performed on all cases of Hughes repair of the lower eyelid performed over a 10-year period (1995 to 2005) to identify patients who required a secondary tarsoconjunctival flap repair. In two cases of tumour recurrence at the lower eyelid margin, an eyelid-sharing, two-stage tarsoconjunctival flap repair of the lower eyelid using the remaining 4 mm of marginal tarsus that was spared in the original Hughes procedure was performed. Case reports of these 2 patients are presented together with their postoperative results. RESULTS: The incidence rate of the described secondary tarsoconjunctival flap procedure was 2 cases per 240 cases (0.8%) of primary Hughes procedure performed over a 10-year period. The postoperative courses in the 2 patients were uncomplicated with excellent surgical results at 6 months' follow-up. CONCLUSIONS: The secondary tarsoconjunctival flap advancement procedure is an effective reconstructive technique for the repair of the full-thickness lower eyelid defect in an eyelid that has already undergone a previous Hughes repair.

Orbital cellulitis as a postoperative complication of sub-Tenon anaesthesia in cataract surgery.

Orbital cellulitis is a rarely reported sight-threatening complication of sub-Tenon anaesthesia. We report a case of orbital cellulitis in a patient who had received sub-Tenon anaesthesia for routine cataract surgery. We discuss the potential under-reporting of complications of sub-Tenon anaesthesia that had a delayed presentation, and the possible association between the use of hyaluronidase in the anaesthetic mixture and orbital cellulitis following sub-Tenon anaesthesia.

Clinical features and management of tumors affecting the lacrimal drainage apparatus.

PURPOSE: To report the clinical features of a series of patients with lacrimal drainage apparatus tumors and present guidelines for management based on histopathology. METHODS: A non-comparative retrospective chart review of the clinical, imaging, and pathologic findings of 37 patients presenting to four regional orbital surgery departments with tumors affecting the lacrimal drainage apparatus between 1990 and 2004. RESULTS: There were 37 patients, of whom 62% were male. The mean age at referral was 54 years. Epiphora, a palpable mass, and dacryocystitis were the most common presentations. Two thirds of the tumors were epithelial, with carcinomas being the most frequent (38%), followed by papillomas (27%). Lymphomas were the most common nonepithelial malignancy (30%). Epithelial tumors were more common in men (87%), whereas lymphomas were more common in women (57%). Treatment modalities included surgery, in addition to radiotherapy and/or chemotherapy and immunotherapy. Mean follow-up was 38 months. Thirty-three patients (89%) remain alive without evidence of disease and 4 patients died of recurrence and/or metastases. CONCLUSIONS: Lacrimal drainage apparatus tumors require careful initial management to ensure adequate local and systemic disease control. Atypical mucosa encountered during dacryocystorhinostomy should be biopsied and small papillomas or pedunculated tumors excised and analyzed with frozen sections. If a diffuse or infiltrative mass is encountered, it should be biopsied and managed on the basis of histopathology and extent of disease. Lymphomas should be treated according to protocols, whereas noninvasive carcinoma and extensive papillomas require complete excision of the system. Invasive disease requires en bloc excision. Long-term follow-up is essential for early detection of recurrence.

Enucleation versus evisceration.

The choice of enucleation and evisceration for removal of an eye remains controversial in certain circumstances. An international panel was asked to give their surgical management of two clinical cases that require either enucleation or evisceration. Case one follows multiple vitreoretinal procedures, and the risk of sympathetic ophthalmia is considered in the surgical management. Case two has had postoperative endophthalmitis, and the possibility of implant infection following insertion of an orbital implant with evisceration and enucleation is discussed.

Orbital and periorbital microcystic adnexal carcinoma.

PURPOSE: To review the clinical and histopathologic features of patients with microcystic adnexal carcinoma of the orbital and periorbital tissues. METHODS: This study was designed as a noncomparative interventional case series of three patients seen and treated at two oculoplastic surgical departments and a review of the literature. Clinical presentation, histopathologic findings, outcome of surgery, and clinical recurrence are presented. RESULTS: Three patients underwent surgery for lesions temporal to the lateral canthus. All had initial histopathology diagnosed as squamous cell carcinoma but were subsequently diagnosed as microcystic adnexal carcinoma after clinical recurrence. All patients had multiple recurrences and extensive perineural spread. Two patients required radiotherapy. CONCLUSIONS: Review of the literature shows that microcystic adnexal carcinoma affecting the orbital and periorbital tissues is difficult to differentiate clinically and microscopically from other conditions including squamous cell carcinoma. Eighty-one percent of cases that have histopathology checked in the initial assessment are still misdiagnosed. A high degree of suspicion is necessary if the lesion extends beyond an apparently adequate surgical margin, multiple recurrences occur, or superficial perineural spread is present. Mohs technique is the treatment of choice and may be improved if paraffin sections or immunohistocytochemistry are performed. Wide excision should be carried out once clear margins are obtained. Radiotherapy may be necessary in cases with multiple recurrences.

Malignant fibrous histiocytoma of the eyelid: differential diagnosis and management.

PURPOSE: Malignant fibrous histiocytoma (MFH) is a pleomorphic soft tissue sarcoma that occurs rarely in the periocular region. The purpose of this study was to present a case series of periocular MFH and to discuss the differential diagnosis and management. METHODS: This is a retrospective case review of patients diagnosed with periocular MFH from tertiary hospitals and private practices. RESULTS: Four patients, two women and two men with periocular MFH, had a mean age of 81 years (range, 72 to 85 years). All tumors were <5 cm in diameter and of storiform pleomorphic histologic subtype. One was located deep and 3 presented superficially. The initial diagnoses were sarcomatoid carcinoma, atypical fibroxanthoma, and leiomyosarcoma that were subsequently reclassified as MFH. One patient had a frozen section, 1 had fast track paraffin section margin control, and 2 had no frozen section margins at the initial excisions. Histology showed 2 negative margins, 1 close margin, and 1 positive margin that were cleared with wide local excision. Local recurrence occurred in 3 cases, and of these, 1 required orbital exenteration. Adjuvant radiotherapy was given to 3 patients. One had regional node metastases. There were no distant metastases or tumor-related deaths. Mean follow-up period was 36 +/- 35 months (median, 30; range, 1 to 84 months). CONCLUSIONS: This study highlights the difficulties in the clinicopathologic diagnosis of periocular MFH and in particular the distinction of more superficial tumors from atypical fibroxanthoma. The mainstay of treatment is complete surgical excision with wide margins, and consideration should be given to histologic margin control in addition to adjuvant radiotherapy.

The cutaneomarginal eyelid graft.

PURPOSE: To assess the effectiveness of the cutaneomarginal graft. METHODS: The cutaneomarginal graft consists of eyelid margin tissue with anterior lamella skin. The tarsal plate is resected following harvesting of a wedge of eyelid tissue. The graft is used to repair eyelid defects following excision of neoplasms which spares the posterior lamella. The graft is principally used at the lateral most part of the lower eyelid following Mohs surgery. A case series of five patients are reviewed to assess the effectiveness of the technique. RESULTS: All patients achieved a satisfactory result following grafting and there were no donor site complications. CONCLUSIONS: The cutaneomarginal graft is an effective means of repair, although with limited indications.

'Ancient' schwannoma of the orbit.

A 65-year-old woman presented with a 3 month history of right eye discomfort and protrusion. Examination revealed right proptosis with hypoglobus and diplopia in extremes of upgaze. Computed tomographic scanning revealed a large extraconal mass in the superotemporal orbit. The mass was excised through an extended superior skin crease incision. Histopathology revealed a benign tumour of Schwann cell origin showing advanced cystic degeneration, the so-called 'ancient schwannoma'. The authors could find only two previously reported cases of such tumours arising in the orbit.

Orbital lymphoid tumour located within an extraocular muscle.

A rare case of an orbital lymphoid tumour within an extraocular muscle is presented. The tumour displayed features of a progressive indolent systemic lymphoma. There was no response to a trial of chemotherapy and so local radiotherapy was instituted. At follow up 2 months later there was no evidence of disease.