Orbital tumours encompass a wide range of benign and malignant space occupying lesions that may arise primarily from the orbit or have spread from other sites in the body. They are rare with an incidence of 1 in every 100 000 and may lead to devastating complications of which mechanical compression causing optic neuropathy is the most important.1 Multiple orbital tumours of the same orbit are even rarer with most reported cases being benign homologous tumours such as cavernous haemangiomas or myxofibrosarcomas.2 3 A 58-year-old ethnic Han Chinese male presented in December 2021 with a 1-month history of right eye proptosis. He had no clinical sign of optic neuropathy. Computed tomography of the orbit revealed right axial proptosis and two separate lesions in the right orbit. One lesion appeared infiltrative and measured 2.2 × 1.5 × 1.7 cm³ (anteroposterior × transverse × longitudinal) at the extraconal space with retrobulbar extension between the lamina papyracea and the medial rectus. The other was an encapsulated mass with regular border located in the superolateral intraconal region measuring 1.9 × 1.7 × 2.0 cm³ and abutting the optic nerve. Both lesions enhanced mildly with intravenous contrast (Fig 1). Blood results revealed normal thyroid function and immunoglobulin G4 and white blood cell levels. Based on the distinguishing radiological features of each lesion, we performed a two-stage surgery for theranostic reasons. An incisional biopsy of the medial infiltrative lesion was performed first through an anterior orbitotomy via an upper lid skin crease approach. Frozen section of the medial yellow jelly-like mass revealed atypical lymphoid cells with enlarged vesicular nuclei and amphophilic cytoplasm, highly suspicious of lymphoproliferative malignancy. We then performed a complete excision of the vascular encapsulated intraconal lesion using cryotherapy via a lateral orbitotomy. Formal histopathology reports revealed the first medial infiltrative lesion to be consistent with diffuse large B-cell lymphoma with positive immunostaining for CD20, BCL2, BCL6, MUM1, and CMYC1 (Fig 2). The second intraconal lesion was consistent with cavernous haemangioma (Fig 3).

At 4 months post-surgery there was no clinical sign of optic nerve damage and best-corrected visual acuity was 0.8 in the right eye. Positron emission tomography–computed tomography showed a residual hypermetabolic lesion over the medial aspect of the right orbit with no extra orbital lesion. The patient is receiving chemotherapy under the care of our haematology team.

Benign multiple homogenous lesions in the same orbit have been reported. Although multiple solitary fibrous tumours of the same orbit without malignant degeneration have been reported,4 multiple heterogeneous tumours in the same orbit are extremely rare. Ma et al5 reported a case of concurrent schwannoma and cavernous haemangioma in the same orbit of a 54-year-old female. To the best of our knowledge, concurrent benign and malignant lesions of the same orbit have not been reported in the English literature.

All authors contributed to the concept and design of this study, acquisition of data, analysis of data, drafting the manuscript, and critical revision for important intellectual content. All authors had full access to the data, contributed to the study, approved the final version for publication, and take responsibility for its accuracy and integrity.

The authors have no conflicts of interest to disclose.

This study received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

The patient was treated in accordance with the Declaration of Helsinki and provided informed consent for the treatment and consent for publication.

1. Demirci H, Shields CL, Shields JA, Honavar SG, Mercado GJ, Tovilla JC. Orbital tumors in the older adult population. Ophthalmology 2002;109:243-8. Crossref

2. Deng C, Hu W. Multiple cavernous hemangiomas in the orbit: a case report and review of the literature. Medicine (Baltimore) 2020;99:e20670. Crossref

3. Du B, He X, Wang Y, He W. Multiple recurrent myxofibrosarcoma of the orbit: case report and review of the literature. BMC Ophthalmol 2020;20:264. Crossref

4. Griepentrog GJ, Harris GJ, Zambrano EV. Multiply recurrent solitary fibrous tumor of the orbit without malignant degeneration: a 45-year clinicopathologic case study. JAMA Ophthalmol 2013;131:265-7. Crossref

5. Ma M, Su F, Yang X. Multiple heterogeneous tumors in orbit: a case report. Int J Clin Exp Pathol 2019;12:4137-41.

A 65-year-old man with a 1-month history of left upper and lower limb chorea was admitted to the medical ward of our institution in February 2021. Blood tests revealed new-onset diabetes mellitus with markedly elevated fasting glucose level of 28.5 mmol/L. Urinalysis for ketones was negative. Urgent non-contrast computed tomography (CT) of the brain showed subtle hyperdensity at the right putamen without mass effect or surrounding oedema (Fig 1). The patient was started on subcutaneous insulin, and upon normalisation of blood glucose level, his hemichorea subsided without additional antichorea medications. Follow-up non-contrast magnetic resonance imaging of the brain performed 2 months later revealed T1-weighted hyperintensity in the right putamen, without restricted diffusion (Fig 2). Imaging findings were in keeping with non-ketotic hyperglycaemic hemichorea (NHH). The patient was prescribed a biphasic insulin regimen upon discharge.

An 87-year-old man with a 2-day history of left upper limb chorea was hospitalised in April 2022. He had known diabetes mellitus but was noncompliant with oral hypoglycaemic therapy. Blood tests revealed markedly elevated random glucose level of 30.8 mmol/L. Urgent non-contrast CT of the brain showed asymmetric subtle hyperdensity in bilateral putamen and caudate nuclei, more extensive on the right, but without mass effect or surrounding oedema (Fig 3). Findings were compatible with NHH. The patient resumed metformin, with gliclazide and subcutaneous insulin injection added for optimal control. Upon normalisation of blood glucose level, his hemichorea resolved without antichorea medications.

Unilateral or asymmetric basal ganglia hyperdensity in brain CT of patients with focal neurological symptoms can be alarming, and intracerebral haemorrhage may be suspected. Nonetheless NHH, a rare complication of uncontrolled diabetes, should not be overlooked.

Case reports of NHH have been documented as early as 1960.1 Previously reported cases were frequent in Asian elderly women with uncontrolled diabetes. In most patients, unilateral chorea was observed, although bilateral involvement could be present.2 3 4 Of note, the aetiology of hemichorea is diverse, and other causes include infarct, haemorrhage and neoplasm. Imaging of the brain is therefore crucial.

Non-contrast CT of the brain of NHH typically shows subtle hyperdensity in contralateral putamen and/or caudate nucleus, although bilateral involvement is also seen. There will be no mass effect or perilesional oedema, and this differentiates NHH from haemorrhage or tumour.2 3 4

Similarly, magnetic resonance imaging of the brain typically shows corresponding signal changes in striatal regions contralateral to the symptomatic side. There will be T1-weighted hyperintense signal. Differentials of increased basal ganglia T1-weighted signal are diverse. They include toxin-related causes such as methanol poisoning or hepatic-related causes such as acquired hepatocerebral degeneration, but they commonly show bilateral and symmetrical involvement.5 T2-weighted or fluid-attenuated inversion recovery signals can be variable. Of note, restricted diffusion is not expected in NHH,2 3 4 and this differentiates it from acute ischaemic stroke.

The pathophysiology of NHH is not fully understood. Proposed mechanisms include depleted gamma-aminobutyric acid and disrupted blood-brain barrier at the corpus striatum.2 3 4 Recognising this rare complication of uncontrolled diabetes mellitus enables prompt medical intervention. Neurological symptoms of NHH usually show substantial improvement after normalisation of blood glucose level without additional intervention.2 3 4

All authors contributed to the concept and design of the study, acquisition of data, analysis and interpretation of data, drafting of the manuscript, and critical revision of the manuscript for important intellectual content. All authors had full access to the data, contributed to the study, approved the final version for publication, and take responsibility for its accuracy and integrity.

All authors have disclosed no conflicts of interest.

This study received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

The patients were treated in accordance with the Declaration of Helsinki. They provided informed consent for all treatments and procedures, and consent for publication.

1. Bedwell SF. Some observations on hemiballismus. Neurology 1960;10:619-22. Crossref

2. Zheng W, Chen L, Chen JH, et al. Hemichorea associated with non-ketotic hyperglycemia: a case report and literature review. Front Neurol 2020;11:96. Crossref

3. Narayanan S. Hyperglycemia-induced hemiballismus hemichorea: a case report and brief review of the literature. J Emerg Med 2012;43:442-4. Crossref

4. Cherian A, Thomas B, Baheti NN, Chemmanam T, Kesavadas C. Concepts and controversies in nonketotic hyperglycemia-induced hemichorea: further evidence from susceptibility-weighted MR imaging. J Magn Reson Imaging 2009;29:699-703. Crossref

5. Hegde AN, Mohan S, Lath N, Lim CC. Differential diagnosis for bilateral abnormalities of the basal ganglia and thalamus. Radiographics 2011;31:5-30. Crossref

An 84-year-old man was admitted with his third episode over 4 months of extended-spectrum beta-lactamase (ESBL)–producing Escherichia coli bloodstream infection (BSI). He had hypertension, diabetes mellitus, benign prostatic hyperplasia, Parkinson’s disease, ischaemic heart disease, and severe aortic stenosis treated with transcutaneous aortic valvular implantation. His most recent glycohaemoglobin level was 6.4% and he was on a controlled diet. He was not on long-term steroids. His two initial infections (presenting only with fever) had each been managed with a 2-week course of intravenous carbapenem and consequent normalisation of inflammatory markers, white blood cell (WBC) count and bacterial clearance on blood culture. Repeated urine culture and liver biochemistry were unremarkable. Transthoracic echocardiogram showed no evidence of vegetation.

The patient was readmitted with a 1-day history of fever with blood culture showing ESBL-producing E coli (refer to the Table for antibiogram) characterised by leukocytosis (WBC count=20×10⁹/L). Systemic review revealed no localising signs or symptoms. We administered 14 days of intravenous meropenem with rapid defervescence and normalisation of WBC. As a deep-seated infection was suspected, whole-body gallium scan was performed and showed intense uptake over the prostate (Fig a). We decided to treat his chronic prostatitis with a prolonged regimen of oral fosfomycin (3 g daily for 1 week, then 3 g every 48 hours for 6 weeks). The patient tolerated fosfomycin without adverse effects (eg, diarrhoea) and remained free of reinfection 3 months after discharge; interval gallium scan showed almost complete resolution of uptake (Fig b).

The primary site of ESBL-producing E coli BSI is predominantly the urinary tract, but may include intra-abdominal infections (eg, pyogenic liver abscess and psoas abscess).1 Typical prostatitis is diagnosed by a 10-time higher bacterial load in expressed prostatic fluid or urine sample collected after prostatic massage than that in a urine sample without prostatic massage.1 Our patient was asymptomatic with a negative urine culture; diagnosis was incidental on gallium scan, confirming its elusiveness. Once diagnosed, chronic prostatitis requires prolonged treatment for 4 to 6 weeks with an appropriate antibiotic.1

Extended-spectrum beta-lactamase–producing E coli arising from prostatitis has significant treatment implications. Few oral antibiotics can adequately penetrate the prostate to be clinically effective. They include fluoroquinolones, trimethoprim/sulfamethoxazole, and fosfomycin.2 3 Fosfomycin has a high clinical success rate and avoids the cardiac and musculoskeletal toxicities traditionally associated with fluoroquinolones.2 In our centre, it has a striking sensitivity of 97%.4

This report highlights three key points. First, chronic bacterial prostatitis should be considered in occult recurrent ESBL-producing Enterobacteriaceae BSI. Second, oral fosfomycin is an excellent choice for ESBL-producing E coli. Third, early stepdown from intravenous to oral antibiotics is effective in real life and validates historical retrospective studies.2 3 Early outpatient management is a pragmatic approach that is especially important within the current context of the coronavirus disease 2019 pandemic where healthcare facilities have been often overwhelmed. Gallium scan or positron emission tomography should be considered for patients with occult infection to determine its origin.5

All authors contributed to the concept or design, acquisition of data, analysis or interpretation of data, drafting of the manuscript, and critical revision of the manuscript for important intellectual content.

All authors had full access to the data, contributed to the study, approved the final version for publication, and take responsibility for its accuracy and integrity.

All authors declared no conflicts of interest.

This study received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

The patient was treated in accordance with the Declaration of Helsinki and provided informed consent for the treatment/ procedures, and consent for publication.

1. Zhanel GG, Zhanel MA, Karlowsky JA. Oral fosfomycin for the treatment of acute and chronic bacterial prostatitis caused by multidrug-resistant Escherichia coli. Can J Infect Dis Med Microbiol 2018;2018:1404813. Crossref

2. Tamma PD, Conley AT, Cosgrove SE, et al. Association of 30-day mortality with oral step-down vs continued intravenous therapy in patients hospitalized with Enterobacteriaceae bacteremia. JAMA Intern Med 2019;179:316-23. Crossref

3. Kwan AC, Beahm NP. Fosfomycin for bacterial prostatitis: a review. Int J Antimicrob Agents 2020;56:106106. Crossref

4. Ho PL, Chan J, Lo WU, et al. Prevalence and molecular epidemiology of plasmid-mediated fosfomycin resistance genes among blood and urinary Escherichia coli isolates. J Med Microbiol 2013;62:1707-13. Crossref

5. Lin KH, Chen YS, Hu G, Tsay DG, Peng NJ. Chronic bacterial prostatitis detected by FDG PET/CT in a patient presented with fever of unknown origin. Clin Nucl Med 2010;35:894-5. Crossref

A previously healthy 4-year-old girl presented with a 4-week history of acute-onset visual disturbance. She was initially prescribed glasses for severe astigmatism but vision did not improve. It was later noted by her parents that her eyes could look only to the left side. She was then admitted to hospital. There was no history of headache, photophobia or vomiting but she exhibited drooling from the right side of her mouth and unsteady gait. She was afebrile and all other vital signs were normal with Glasgow Coma Scale score of 15. Physical examination revealed bilateral fixed left lateral gaze and right lower-motor-neuron seventh cranial nerve palsy. When asked to look to her right, she needed to compensate by head turning. Pupils were 3 mm equal and reactive to light. Ear canals were normal and there was no skin rash. Computed tomography scan of the brain showed a multilobulated mixed hyperdense and hypodense lesion at the dorsal pons with compression of the fourth ventricle. Magnetic resonance imaging (MRI) of the brain revealed bleeding from a tumour at the right dorsal pons (Fig 1); overall picture suggested multiple cavernoma (Fig 2). Neurosurgical decompression and stereotactic excision of a brainstem lesion was performed via suboccipital craniotomy. Histology showed features of a vascular lesion in keeping with cavernous haemangioma. Intracranial pressure was monitored via an external ventricular drain and remained normal postoperatively. Postoperative neurological examination showed bilateral fixed left lateral gaze, right lower-motor-neuron seventh cranial nerve palsy, and contralateral weakness of limbs. Genetic testing demonstrated a pathogenic mutation [heterozygous KRIT1 c.690C>A p.(Tyr230*)] for familial cerebral cavernous malformation (CM) syndrome. Her family was referred for further genetic testing and counselling.

Acute-onset gaze disturbance in children is alarming and may signify serious cerebral abnormality. Almost all conjugate gaze palsies originate from a lesion in the midbrain or pons. These lesions can be caused by vascular or oncological space occupying lesions.

Cavernous malformations, also known as cavernous haemangiomas, are a type of benign, congenital malformation in which a cluster of dilated thin-walled capillaries form a characteristic ‘mulberry’ lesion with engorged purplish colour.1 They can be sporadic or inherited with an autosomal dominant pattern and incomplete penetrance, and can present as solitary or multiple lesions. Unlike capillary haemangiomas, CMs can be life-threatening and do not tend to regress. In all, 25% of cerebral CMs are infratentorial. They have a bleeding rate of 2% to 3% per year and recurrent bleeding rate of >20%. Due to the close proximity to multiple brainstem nuclei and fibre tracts, progressive neurological decline is observed in 39% patients with infratentorial CMs.2 Magnetic resonance imaging is the modality of choice, and susceptibility weighted imaging is the most sensitive means to detect blood products thus key to diagnosing cerebral CMs. Evolving blood products inside a CM appear as variable image intensities and give rise to the typical ‘popcorn’ appearance.3 Since MRI appearance is usually pathognomonic, biopsy is rarely needed. Angiography is indicated only if MRI cannot exclude arteriovenous malformation. Genetic testing should be arranged to screen for familial cerebral CM. Treatment approach depends on the site, size, symptoms, and history of haemorrhage.4 Indications for surgical resection of a cerebral CM include intracranial haemorrhage and epilepsy. Options include conventional surgery or stereotactic radiosurgery.4 5

All authors contributed to the concept or design, acquisition of data, analysis or interpretation of data, drafting of the manuscript, and critical revision of the manuscript for important intellectual content.

All authors had full access to the data, contributed to the study, approved the final version for publication, and take responsibility for its accuracy and integrity.

As an editor of the journal, KL Hon was not involved in the peer review process. Other authors have disclosed no conflicts of interest.

This study received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

Ethics approval for this study was obtained from Hong Kong Children’s Hospital Ethics Committee, Hong Kong (Ref No.: HKCH REC 2019 009). The patient was treated in accordance with the tenets of the Declaration of Helsinki. Written consent for publication has been obtained from the patient’s parent.

1. Awad IA, Polster SP. Cavernous angiomas: deconstructing a neurosurgical disease. J Neurosurg 2019;131:1-13. Crossref

2. Fritschi JA, Reulen HJ, Spetzler RF, Zabramski JM. Cavernous malformations of the brain stem. a review of 139 cases. Acta Neurochir (Wien) 1994;130:35-46. Crossref

3. Lehnhardt FG, von Smekal U, Rückriem B, et al. Value of gradient-echo magnetic resonance-imaging in the diagnosis of familial cerebral cavernous malformation. Arch Neurol 2005;62:653-8. Crossref

4. Poorthuis MH, Klijn CJ, Algra A, Rinkel GJ, Al-Shahi Salman R. Treatment of cerebral cavernous malformations: a systematic review and meta-regression analysis. J Neurol Neurosurg Psychiatry 2014;85:1319-23. Crossref

5. Wang CC, Liu A, Zhang JT, Sun B, Zhao YL. Surgical management of brain-stem cavernous malformations: report of 137 cases. Surg Neurol 2003;59:444-54. Crossref

Eczema is the most prevalent childhood atopic illness routinely encountered by health professionals providing care to children.1 Some infants become critically ill and present to the paediatric intensive care unit (PICU) with this seemingly trivial condition.2 This anonymised (no clinical and laboratory data are presented) case of severe eczema and multi-organ dysfunction illustrates the extent of multi-organ involvement in eczema and the medical and psychosocial issues associated with the disease.1 3

A 6-month-old boy with eczema and septic shock presented to the emergency department. He was treated with fluid resuscitation and subsequently admitted to a PICU. Derangement of multiple organ functions were identified along with failure to thrive and poor development (Table, Figs 1 and 2). Dobutamine and broad-spectrum antibiotics were prescribed. Although his condition improved rapidly, it became apparent that his parents did not trust Western medicine and were reluctant to use emollient and topical steroid to manage their child’s eczema. The mother frankly admitted that since the development of eczema at 2 months of age, she had been giving the infant complementary herbal medicine remedies. She was also taking herbal medicine whilst breastfeeding so that her child would indirectly receive the more ‘effective’ herbal medicine. The parents were interviewed by healthcare workers from various disciplines but they remained resolute in their beliefs and phobias about Western medicine. The child was discharged from PICU after the acute medical issues were resolved. Extensive investigations have been performed and apart from persistent peripheral eosinophilia, slightly low immunoglobulin M level, borderline CD4:CD8 ratio and borderline low zinc level, all other investigations including metabolic workup remain unremarkable to date. Further paediatric dermatology, genetics and integrative medicine follow-ups have been arranged.

Our reported case is probably the youngest patient with critical ‘status eczematicus’ to survive. All growth parameters were compromised (Fig 1). Children with severe eczema rarely require admission to an ICU and there are very few such cases in the literature. Mortality due to eczema is rare, but we have previously reported a tragic case of an infant with eczema who died of group B streptococcus septicaemia and malnutrition despite expensive dietary supplements.2 Eczema is a chronic condition that can significantly affect a child’s quality of life as well as that of their family if it is not well controlled due to the potentially significant psychological toll.4 Although ICU is not an ideal setting to manage a family with multiple phobias, the child must first be stabilised, and other differential diagnoses of acute skin failure ruled out.2

Acute treatment of eczema is straightforward but long-term maintenance treatment is always challenging. Topical medications should be considered to prevent exacerbations and therapy should be proactive. Steroid phobia is prevalent and often leads to non-compliance.1

Recommendations about dietary avoidance should be specific and given only in confirmed cases of food allergy.1 The use of traditional and proprietary topical and herbal medicine is popular across many countries in Asia.1 Anxious food-avoiding parents may purchase multi-vitamin supplements, prebiotics, probiotic or symbiotics that claim to be effective. In an extreme case, death was reported to have a secondary association with extreme dietary practice.3 Physicians must be tactful when counselling these anxious parents who are steroid phobic and mistrusting of modern medicine. Indirect administration of herbal medicine to an infant through breastfeeding is not advocated even in the practice of integration medicine.

Management of eczema can sometimes be challenging. Apart from strong parental beliefs, cultural differences might also play a role in compliance with a prescribed treatment plan or a tendency to seek alternative therapies instead. These effects are likely to be underestimated in our community, especially in the primary care setting where consultation time might be limited. Physicians should endeavour to spend more time exploring parental beliefs. A practical solution may be to use an objective self/parental assessment to aid eczema control assessment, eg, ‘Traffic Light Control’ self-assessment system (Fig 3) and quality of life assessment, eg, Children’s Dermatology Life Quality Index.5 An ‘eczema action plan’ can also be given to parents to remind them of the prescribed eczema treatment. The treatment plan should be straightforward and easy to follow, especially if they perceive a worsening of eczema between clinic visits.5 A multidisciplinary and perhaps integrative medicine approach should be adopted where possible to manage patients and families with eczema, and education about the disease should be individualised to improve patient outcomes.

Healthcare providers must be aware of the mortality and morbidity associated with recalcitrant eczema and ‘status eczematicus’. These tragic cases of ‘status eczematicus’ serve to remind us of the grave consequences if eczema is inappropriately managed.

All authors contributed to the concept or design, acquisition of data, analysis or interpretation of data, drafting of the manuscript, and critical revision of the manuscript for important intellectual content.

All authors had full access to the data, contributed to the study, approved the final version for publication, and take responsibility for its accuracy and integrity.

As an editor of the journal, KL Hon was not involved in the peer review process. Other authors have disclosed no conflicts of interest.

This study received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

The patient was treated in accordance with the tenets of the Declaration of Helsinki. Ethics approval for publication of patient information in the paediatric intensive care unit was obtained from the Hong Kong Children’s Hospital Research Ethics Committee (Ref No.: HKCH-REC-2019–0011).

1. Hon KL, Leong KF, Leung TN, Leung AK. Dismissing the fallacies of childhood eczema management: Case scenarios and an overview of best practices. Drugs Context 2018;7:212547. Crossref

2. Hon KL, Nip SY, Cheung KL. A tragic case of atopic eczema: malnutrition and infections despite multivitamins and supplements. Iran J Allergy Asthma Immunol 2012;11:267-70.

3. Hon KL, Kam WY, Leung TF, et al. Steroid fears in children with eczema. Acta Paediatr 2006;95:1451-5. Crossref

4. Hon KL, Kung JS, Wang M, Pong NH, Li AM, Leung TF. Clinical scores of sleep loss and itch, and antihistamine and topical corticosteroid usage for childhood eczema. Br J Dermatol 2016;175:1076-8. Crossref

5. Lam PH, Hon KL, Leung KK, Leong KF, Li CK, Leung TF. Self-perceived disease control in childhood eczema. J Dermatolog Treat 2022;33:1459-646. Crossref