Doxycycline sclerotherapy in patients with periorbital lymphatic malformation: A case series and literature review.

PURPOSE: To describe and report the outcomes of doxycycline sclerotherapy in patients with periorbital lymphatic malformations(LMs). BASIC PROCEDURES: A retrospective review of consecutive patients diagnosed with periorbital LMs and who received doxycycline sclerotherapy at Hong Kong Eye Hospital and Queen Elizabeth Hospital, Hong Kong between January 2016 and June 2022. Doxycycline was prepared with a concentration of 100 mg diluted in 10 mL water for injection. A 23-gauge needle aiming at the center of the macrocyst was used to aspirate fluid from the lesion; this was then followed by an intralesional injection of 0.5 to 2 ml of doxycycline depending on the size of the cavity. MAIN FINDINGS: A total of eight patients(six females) were included in this study. All of them received doxycycline sclerotherapy for periorbital LMs(five extraconal, three intraconal). The median age for receiving sclerotherapy was 29 years old. Seven patients had macrocystic LMs, and one had mixed macro- and microcystic LM. Two of the LMs had venous components radiologically. The average number of sclerotherapy treatment in one patient was 1.4 ± 0.7times. Seven of the eight patients had excellent response radiologically or clinically. One patient showed a satisfactory response after three cycles of sclerotherapy. No recurrence was experienced at median follow-up of 14 months. None of the patients experienced visual threatening or systemic complication. PRINCIPLE CONCLUSIONS: Our preliminary experience with doxycycline sclerotherapy has shown encouraging results for the treatment of macrocystic or mixed-type periorbital LMs, with a favourable safety profile. Further clinical trials with longer follow-ups are warranted on this topic.

Atypical mycobacterium infection following upper eyelid Müller's muscle-conjunctival resection - case report.

Periorbital non-tuberculous mycobacterium (NTM) infections are uncommon. To the best of our knowledge, NTM infection as a complication following Müller's muscle-conjunctival resection (MMCR) surgery has not been reported before. We report a case of left upper lid M. Chelonae infection following MMCR surgery. A 61-year-old lady presented with left upper lid swelling and nodular mass 4 weeks after bilateral MMCR surgery for aponeurotic ptosis. Past medical and ocular history include systemic lupus erythematosus (SLE), chronic hepatitis B infection, bilateral cataract operation done 14 years ago and right eye Fuch's dystrophy with Descemet stripping automated endothelial keratoplasty done 3 years ago. She was initially treated with topical and oral antibiotics, as well as repeated incision and curettage and intralesional steroid injection with limited improvement. Seven months post-MMCR, repeated biopsy and nodule debulking were performed. Biopsy revealed granulomatous inflammation with mycobacterial infection and PCR identified M. Chelonae. A total of 6 months course of combination systemic antibiotics were given, with good response. Limited blepharoplasty with repeat nodular excision was performed 15 months after the initial MMCR surgery, and biopsy culture and PCR were both negative. No relapse of symptoms was noted and good lid height was maintained at 30 months of follow-up. Management of periorbital NTM infections can be challenging. Clinicians should consider early diagnostic workup with mycobacterial culture and PCR in suspicious cases, followed by prompt initiation of empiric treatment with systemic macrolides. A combination of surgical excision of nodules and prolonged systemic antimicrobial treatment is needed for complete organism eradication.

Giant Fornix Syndrome: How Much Conjunctiva Should Be Resected?

PURPOSE: To characterize clinical profiles of Chinese patients with giant fornix syndrome (GFS), compare surgical outcomes with amount of Müller's muscle-conjunctival resection (MMCR), and elicit risk factors for those who have prolonged recovery after MMCR. METHODS: Retrospective, observational, interventional cohort study on GFS eyes. Two treatment groups were established: limited MMCR-as defined by 8 mm or less resection; maximal MMCR-as defined by 10-12 mm resection. Good responders were defined as eyes exhibiting disease resolution within 3 months after surgery. Primary outcome was disease resolution, secondary outcome was ptosis improvement. RESULTS: Mean age was 81.9 years old (range, 76-89), with 6 (75%) females and 2 (25%) males. All 10 eyes presented with discharge, partial ptosis, and conjunctival injection. In the limited MMCR group, time to symptom resolution was longer at 5.56 months, while maximal MMCR group was 2.02 months (p = 0.004). Limited MMCR group also had lower primary surgical success and required additional surgery compared with maximal MMCR group (p = 0.008). At mean follow up of 34.4 months (range, 11-65 months), all eyes achieved disease resolution, no recurrence, and ptosis improvement. CONCLUSIONS: In the largest series on Chinese eyes with GFS to date, GFS is mainly a disease in elderly females. Maximal MMCR has a higher rate of surgical success with no additional complications. For those who underwent MMCR, additional treatment such as topical steroids and fortified antibiotics do not affect time to recovery. These findings may help ophthalmologists consider maximal MMCR as a definitive surgical treatment in GFS eyes.

Transnasal Endoscopic Sclerotherapy for Orbital Apex Lymphatic Malformation.

Orbital lymphatic malformations are benign, slowly progressive vascular malformations. Management of these malformations is challenging due to their infiltrative and diffuse nature. The authors present a case with orbital apex lymphatic malformation treated with transnasal endoscopic sclerotherapy.

Extraocular silicone oil migration to orbit and retrolaminar region: case report and systematic review.

BACKGROUND: Silicone oil insertion is a common modality in vitreoretinal surgeries. The purpose of this study is to conduct a systematic review to summarize the uncommon complication of extraocular silicone oil migration. METHODS: Following the PRISMA guidelines, a systematic review of the literature was performed on January 11, 2020, using PubMed and EMBASE with the following terms: "silicone oil," "eye," and "migrat*." RESULTS: A total of 69 patients-68 patients from 59 articles and one case from our institution-were included in the final analysis. The median age was 54 years (range, 9-92) and 40 patients (57.9%) were men. Orbital migration was reported in 34 patients, and retrolaminar migration (including optic nerve, optic chiasm, suprasellar, subarachnoid space, intraventricular spaces) was reported in 35 patients. Orbital migration group had more aphakics (p = 0.007), implanted glaucoma drainage device (p = 0.005), scleral buckle (p = 0.000), history of trauma-related indications for pars plana vitrectomy (p = 0.000), shorter silicone oil endotamponade time (p = 0.008), more symptomatic (p = 0.000), and requiring surgical intervention (p = 0.000). Retrolaminar migration group had older patients (p = 0.016) and more diabetics (p = 0.041). CONCLUSION: Systematic review sheds light on plausible risk factors on site of silicone oil migration. Majority of orbital cases are symptomatic and require intervention while retrolaminar cases are incidental and can be managed conservatively. Awareness of this complication can help guide clinicians predict which patients would likely need surgical intervention. Graphical abstract.

Medical and sclerosing agents in the treatment of orbital lymphatic malformations: what's new?

PURPOSE OF REVIEW: Currently, there is no ideal management for orbital lymphatic malformations. Significant advances have been made since the discovery of new agents in the treatment. The purpose of this manuscript is to review the recent evidence on new sclerotherapy agents and systemic medications. RECENT FINDINGS: Traditional sclerosants are OK-432, sodium tetradecyl sulphate and ethanol. More recent developments are the use of doxycycline, bleomycin, and pingyangmycin. Sirolimus as a systemic medication has revolutionized the medical management of lymphatic malformations. Other oral drugs such as propranolol and sildenafil are controversial. Future treatment involves targeting lymphangiogenic pathways including inhibition of vascular endothelial growth factors and the phosphatidylinositol 4,5-bisphosphate 3-kinase catalytic subunit. SUMMARY: The development of new agents allows multimodal management either as monotherapy or combined therapy to achieve better outcomes in this difficult to manage disease.

The practice patterns in the management of sebaceous carcinoma of the eyelid in the Asia Pacific region.

PURPOSE: To determine the practice patterns of ophthalmic plastic surgeons regarding the management of eyelid sebaceous carcinoma (SC). METHODS: An electronic survey was distributed to oculoplastic surgical colleagues in the Asia Pacific region requesting clinical information and treatment approaches to SC. RESULTS: The responses from 192 respondents from the Asia Pacific region was included and analyzed in this study. For initial diagnosis, most surgeons selected incisional biopsy (55%), followed by complete excision (35%). Initial workup was mainly by palpation of lymph nodes, chest X-ray, and computerized tomography scan (CT-scan) of the orbit. Conjunctival map biopsy was done in selected cases. Sentinel lymph node biopsy (SLNB) was done mainly for tumors larger than 10 mm. Management was mainly by surgical excision (5 mm margin) combined with adjuvant therapy in some cases, with radiotherapy being the most common. Margin status was determined most frequently by frozen section as evaluated by the pathologist (57%) followed by Mohs micrographic surgery (18%). Surveillance was based mainly on physical examination alone. CONCLUSION: The Asia Pacific oculoplastic surgeons prefer incisional biopsy for lesions suspicious of SC prior to definitive surgery. This is in contrast to survey results previously reported in other populations. Frozen section control (done by an oculoplastic surgeon with pathology support) is most commonly used for margin control and conjunctival map biopsies are done only in selected cases. Despite the potential benefits of SLNB, access and expertise in this area is currently lacking in the Asia Pacific region.

Unilateral Eyelid Swelling as a Sign of Antimelanoma Differentiation-Associated Gene 5 (Anti-MDA5)-Antibody-Positive Dermatomyositis.

The authors describe a 40-year-old woman who presented with unilateral eyelid swelling, and later on developed heliotrope rash, extensive oral and cutaneous ulcers, and muscle weakness. She was diagnosed with dermatomyositis associated with antimelanoma differentiation-associated gene 5 antibody. Even after combination of immunosupressants including a corticosteroid, tacrolimus, and rituximab, her eyelid swelling and oral ulcers progressively worsened. Administration of loading and then monthly intravenous immunoglobulin resulted in clinical remission, suggesting that intravenous immunoglobulin could be a useful remedy in cases refractory to conventional immunosuppressive agents, especially those with antimelanoma differentiation-associated gene 5 antibody-dermatomyositis.

14-year case series of eyelid sebaceous gland carcinoma in Chinese patients and review of management.

AIMS: Sebaceous gland carcinoma (SGC) of the eyelid is a rare but potentially deadly cancer. The purpose of this study was to determine whether the American Joint Committee on Cancer (AJCC) 7th Edition T category for SGC correlated with metastasis and survival in the Chinese population. METHODS: This was a retrospective, single-centre cohort study. Patients with surgically resected eyelid SGC between January 2001 and May 2015 at the Hong Kong Eye Hospital were reviewed. Tumours were staged using the AJCC criteria. The main outcome measures included local recurrence, metastasis and death. Disease-free survival (DFS) was measured from the completion of treatment; overall survival was measured from the date of initial diagnosis. RESULTS: The study included 22 Chinese patients with a mean age of 65.4 years. The majority presented as a nodular lesion (91%) with 12 eyes (54.5%) initially misdiagnosed and a mean presentation time of 1 year. It was found that those with AJCC stage T2b or higher were significantly associated with lymph node metastasis (P=0.002) when compared with those with stage T2a. Older age at diagnosis (P=0.035) and no misdiagnosis (P=0.025) were associated with shorter DFS. Those with stage 3a or higher were associated with shorter DFS (P=0.007) and overall survival (P=0.024). CONCLUSION: Similar to previous reports, in this Chinese cohort, AJCC staging for SGC correlated with lymph node metastasis, DFS and overall survival. Those with stage 2b or higher on presentation will need closer surveillance for lymph node metastasis and may benefit from sentinel lymph node biopsy.

Digital ischaemia: a rare but severe complication of jellyfish sting.

We report a case of digital ischaemia in a 31-year-old man who presented with sudden hand numbness, swelling, and cyanosis 4 days after a jellyfish sting. This is a rare complication of jellyfish sting, characterised by a delayed but rapid downhill course. Despite serial monitoring with prompt fasciotomy and repeated debridement, he developed progressive ischaemia in multiple digits with gangrenous change. He subsequently underwent major reconstructive surgery and aggressive rehabilitation. Although jellyfish stings are not uncommon, no severe jellyfish envenomation has been reported in the past in Hong Kong and there has not been any consensus on the management of such injuries. This is the first local case report of jellyfish sting leading to serious hand complications. This case revealed that patients who sustain a jellyfish sting deserve particular attention to facilitate early detection of complications and implementation of therapy.

Fever as a predictor of doctor shopping in the paediatric population.

OBJECTIVES: To estimate prevalence and assess factors associated with doctor shopping among caregivers of children acutely admitted to a hospital in Hong Kong, and examine the reasons for such behaviour and caregivers' awareness of its possible dangers. DESIGN: Cross-sectional study with face-to-face surveys. SETTING: A paediatric unit in a teaching hospital in Hong Kong. PARTICIPANTS: Caregivers of children admitted to acute paediatric wards between April and July 2011. MAIN OUTCOME MEASURES; Socio-demographic characteristics of the interviewee, personal history and clinical data of the patient, presence of doctor shopping (consulting more than one doctor for medical advice without referral) for each episode, the reasons behind such behaviour, and awareness of potential dangers. Data retrieved were analysed to estimate the prevalence and logistic regression was used to assess factors associated with doctor shopping. RESULTS: In all, 649 such patients were admitted into hospital during the study period, of which 336 were recruited, with about a half being absent or given home leave. Thirty-four patients were excluded due to absent caregivers or refusal, and 302 were included in the study. More than half (79.5%) were female and the caregivers' monthly household incomes were between HK$10 001 and HK$15 000 (21.2%), similar to the median household income in Hong Kong. The prevalence of doctor shopping was 53%. The only significant clinical parameter associated with doctor shopping was presence of fever (odds ratio=2.4; 95% confidence interval, 1.4-3.9). Persistence of symptoms was the commonest reason given by interviewees for doctor shopping, and the majority (75.5%) were unaware of the possible dangers of this behaviour. CONCLUSION: Doctor shopping is highly prevalent among caregivers of children with acute paediatric conditions. Most caregivers do not know the potential complications of this behaviour. Further measures should be taken to educate subjects on the associated dangers of this behaviour and the natural course of acute illnesses with fever.