In July 2020, a 35-year-old pregnant woman (weight 70 kg, height 160 cm, body mass index 27.3) at 34+3 weeks of gestation presented to our hospital with upper respiratory tract infection symptoms including dry cough and runny nose for 4 days. The patient had a history of lower segment caesarean section in 2016, with good past health and an unremarkable antenatal history. Reverse transcription polymerase chain reaction analysis of the patient’s deep throat saliva sample was positive for severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). The cycle threshold value was 19.79 indicating a high viral load. She had no fever or shortness of breath at any time.

The patient also presented with a small amount of per-vaginal spotting but had no abdominal pain or leaking sensation. Fetal movements were active. The patient’s blood pressure was 98/62 mm Hg, pulse was 97 bpm, SpO₂ was 97% on room air, and body temperature was 37°C. Fetal ultrasonography was unremarkable with appropriate size for gestational age. The impression was of antepartum haemorrhage of unknown origin and maternal coronavirus disease 2019 (COVID-19) infection.

The patient had normal liver and renal function, clotting profile and chest radiograph, and negative results for influenza A/B and respiratory syncytial virus tests. Other results included: haemoglobin 11.1 g/dL, platelet count 198×10⁹/L, neutrophilia of 73% white blood cell differential count, lymphopenia at 0.9×10⁹/L 16.5% of white blood cell differential count, and C-reactive protein 8 mg/L (ref <5 mg/L).

The patient was admitted to a negative pressure isolation ward, standard practice for airborne infection. A multidisciplinary meeting was held involving obstetricians, anaesthesiologists, paediatricians, microbiologists, infectious disease specialists, and intensivists. The consensus, agreed with the patient, was that an early elective lower segment caesarean section under spinal anaesthesia should be performed the next day.

Routes were isolated and protected for transferring patients between the operating theatre (OT), the isolation ward, and the neonatal intensive care unit, and for transferring healthcare workers from the OT to decontamination shower facilities. Only essential equipment, medications, and consumables were placed inside the OT. Disposable consumables were used whenever possible. Paper records were minimised and sealed.

Preparations were made inside the OT for spinal anaesthesia, and for conversion to general anaesthesia if necessary. Medications for induction and resuscitation were prepared in advance, including propofol, succinylcholine, cisatracurium, phenylephrine, ephedrine, atropine, neostigmine, syntocinon, carbetocin, and tranexamic acid. A video laryngoscope with disposable blades, stylet, elastic gum bougie, endotracheal tubes and size 3 and 4 classic and supreme laryngeal masks were placed under plastic sheet covers.

Staff exposure time was minimised, but this was balanced against an optimal standard of patient care. Only active essential personnel were present in the OT for each stage of surgery. All staff inside the OT wore level 3 personal protective equipment, including fluid-resistant long-sleeve gown, gloves, face shield, and fit-tested N95 masks according to airborne precautions. During spinal anaesthesia, only two anaesthesiologists and an assistant were present in the OT. The anaesthesiologists wore water-resistant sterile gowns during the spinal anaesthesia. Other staff, including the surgeons and midwives, waited in the anteroom entering the OT only when the anaesthesia was complete.

The patient wore a water-resistant surgical mask throughout the transfer and surgery. She required no supplementary oxygen. On entering the OT, her blood pressure was 120/82 mm Hg, pulse 91 bpm, and SpO₂ 99.4% on room air. Anaesthesia was induced by 2.2 mL of 0.5% hyperbaric bupivacaine, 15 μg of fentanyl, and 0.15 mg morphine injected intrathecally via a 25-gauge pencil tip spinal needle in a single attempt, at the L3-L4 level, with sensory block to T5 bilaterally. Phenylephrine (100 μg/mL) infusion at 15 mL/h (0.36 μg/kg/min) was commenced 5 minutes after spinal anaesthesia. A baby girl was delivered 19 minutes after the spinal injection. Total blood loss was 1000 mL, and 2000 mL of Plasma-Lyte A was infused. A phenylephrine infusion was titrated down and stopped on completion of the surgery. The patient’s blood pressure was 116/56 mm Hg and her pulse was 68 bpm. Monitoring continued in the same OT to avoid contamination of the post-anaesthesia care unit.

Postoperative analgesia included oral paracetamol 1 g four times daily and diclofenac sodium sustained release 100 mg daily. The patient was followed up daily. She was satisfied with the overall anaesthetic experience and pain control. The baby had 1-minute and 5-minute Apgar scores of 8. Repeated tests of nasopharyngeal aspiration and throat swab were negative for SARS-CoV-2 infection; however, the patient was nursed in an isolation ward in the neonatal intensive care unit as a precaution.

Samples including amniotic fluid, placental swab, high vaginal swab and breast milk all tested negative for SARS-CoV-2. All personnel directly involved in care of the mother and the baby and those involved in OT decontamination remained symptom free after 14 days of medical surveillance.

The timing of delivery was a major concern for this patient. If the mother’s health had deteriorated before delivery, the use of certain antiviral medications would have caused deranged organ function and affected fetal well-being. Corticosteroid may have harmed the mother.1 2 Use of corticosteroids in preparation for premature labour is thought to cause a worse outcome in patients with COVID-19.3 Fortunately our patient was at >34 weeks’ gestation. Unexpected deterioration of the mother’s health could also result in the need for an unanticipated urgent operative delivery, imposing increased operative risks, as well as increased infectious risks to healthcare workers. In particular, general anaesthesia requires endotracheal intubation that is considered an aerosol-generating procedure. Spinal anaesthesia offered a good alternative to general anaesthesia in a planned setting, as illustrated by a case series from Wuhan, China, of 49 caesarean deliveries with good blood pressure control achieved under spinal anaesthesia.4

Early data suggest that pregnant women do not develop more severe COVID-19.5 In a study of 241 births in the United States, approximately 30% of mothers with COVID-19 became symptomatic, 7.1% required intensive care unit admission, 3.7% required intubation, and 0.7% progressed to a critical condition. The deterioration could be rapid and occurred over a variable time frame.5 Current guidelines suggest that COVID-19 infection by itself is not an indication for early induction of labour or operative delivery, but that the timing of delivery should be determined by obstetric indications.6

This patient presented with antepartum haemorrhage of unknown origin, and minor placental abruption could not be excluded. Any deterioration in placental abruption could rapidly jeopardise the well-being of the fetus and mother. The decision for early elective operative delivery was appropriate in these circumstances.

Concern was expressed about high viral load and infectivity, given the patient’s high cycle threshold value of 19.79. However, there is wide variation in the interpretation of cycle threshold,7 and no evidence that a lower cycle threshold value correlates with worse prognosis.8

This case report confirms that it is possible for a patient with confirmed SARS-CoV-2 infection to safely undergo spinal anaesthesia with maintenance of stable blood pressure.4 Our findings suggest that pregnant women with mild COVID-19 symptoms are little different to healthy pregnant women who undergo spinal anaesthesia for caesarean section.

Overall, this case report demonstrates that with the input of anaesthesiologists, joint clinical decisions and effective communication between relevant specialties, a well-planned and rehearsed routing, and correct use of personal protective equipment, the infectious risks to health care professionals could be minimised while providing an appropriate standard of care to the mother and the baby.

All authors contributed to the concept or design of the study, acquisition of the data, analysis or interpretation of the 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.

The authors declare that they have no conflict of interest.

This case report 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. The patient provided written informed consent for all treatments and procedures.

1. Sanders JM, Monogue ML, Jodlowski TZ, Cutrell JB. Pharmacologic treatments for coronavirus disease 2019 (COVID-19): a review. JAMA 2020;323:1824-36. Crossref

2. Bauer ME, Bernstein K, Dinges E, et al. Obstetric anesthesia during the COVID-19 pandemic. Anesth Analg 2020;131:7-15. Crossref

3. Society for Maternal-Fetal Medicine, Society for Obstetric and Anesthesia and Perinatology. Labor and delivery COVID-19 considerations. Available from: https:// s3.amazonaws.com/cdn.smfm.org/media/2402/SMFM-SOAP_COVID_LD_Considerations_-_revision_6-16-20_PDF.pdf. Accessed 26 Jul 2020.

4. Zhong Q, Liu YY, Luo Q, et al. Spinal anaesthesia for patients with coronavirus disease 2019 and possible transmission rates in anaesthetists: retrospective, single-centre, observational cohort study. Br J Anaesth 2020;124:670-5. Crossref

5. Khoury R, Bernstein PS, Debolt C, et al. Characteristics and outcomes of 241 births to women with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection at five New York City Medical Centers. Obstet Gynecol 2020;136:273-82. Crossref

6. National Institutes of Health. Coronavirus disease 2019 (COVID-19) treatment guidelines. Available from: https://www.covid19treatmentguidelines.nih.gov/. Accessed 25 Jul 2020.

7. Han MS, Byun JH, Cho Y, Rim JH. RT-PCR for SARS-CoV-2: quantitative versus qualitative. Lancet Infect Dis 2021;21:165. Crossref

8. Young BE, Ong SW, Kalimuddin S, et al. Epidemiologic features and clinical course of patients infected with SARS-CoV-2 in Singapore. JAMA 2020;323:1488-94. Crossref

In areas endemic for tuberculosis infections, the presence of infection on imaging and granulomatous inflammation on histology is often sufficient to start antituberculous pharmacotherapy. However, this may not be appropriate in cases of non-tuberculous granulomatous infection. Rarer causes of spinal infection should be considered, especially if the clinical response is suboptimal. We present an unusual case of granulomatous spinal infection caused by Coxiella burnetii.

A 74-year-old man from southern China with no travel history presented with a 3-month history of low back pain in the absence of neurological deficit or constitutional symptoms. Co-morbidities included hypertension, diabetes mellitus, and hyperlipidaemia. There were no symptoms or signs of endocarditis. Initial plain radiographs showed lytic destruction of L4 and erosion of the L4-L5 disc space. Laboratory investigations revealed raised C-reactive protein (CRP) of 27.6 g/dL and erythrocyte sedimentation rate of 32 mm/h. Blood culture, Widal test for Salmonella, HIV testing and acid-fast bacilli growth in sputum and blood were all negative. There was no growth of fungus or brucellosis. Magnetic resonance imaging and computed tomography (CT) revealed L4/5 spondylodiscitis with psoas and paravertebral abscesses (Fig 1), and a mycotic infrarenal abdominal aortic aneurysm measuring 7.3 × 7.6 × 5.7 cm. He underwent endovascular stenting of the aneurysm. A CT-guided aspiration of the psoas abscess was negative on Ziehl–Neelsen and Grocott staining and tuberculous polymerase chain reaction. Histology confirmed granulomatous inflammation. After discussion with the microbiologist, the patient was prescribed intravenous ceftriaxone 2 g per day and metronidazole 1 g per day but switched to ertapenem 1 g per day due to a poor clinical response. These antibiotics were used as empirical therapy. Symptoms subsided and his CRP normalised to <0.35 g/dL. He was discharged home and prescribed lifelong amoxicillin/clavulanic acid 375 mg 3 times daily, with levofloxacin 500 mg once daily for the mycotic aneurysm.

At 6 months after discharge he presented with recurrent back pain along with radicular symptoms. Inflammatory markers were elevated with CRP 3.5 g/dL and erythrocyte sedimentation rate 81 mm/h. The same cultures and serology were negative. Plain radiographs and CT showed increased bony destruction of L4. Magnetic resonance imaging revealed persistent retroperitoneal abscess with progressive spondylodiscitis (Fig 2). A CT-guided drainage of his right psoas abscess yielded negative culture results. Due to the neurological deterioration, an L3-L5 laminectomy with posterior instrumented spinal fusion was performed. Anterior column reconstruction was attempted by the vascular surgeon but scarring prevented safe access to the spinal column without aneurysmal injury. The L4-L5 disc material revealed granulomatous inflammation and methicillin-resistant Staphylococcus aureus. He was treated as co-infection with tuberculosis and methicillin-resistant Staphylococcus aureus, and was given isoniazid 300 mg, rifampicin 450 mg, and ethambutol 800 mg once daily, and linezolid 600 mg every 12 hours. His inflammatory markers normalised but back pain persisted.

Serial radiographs showed implant failure with further destruction of L4 (Fig 3) due to the lack of anterior column support. Revision posterior instrumented fusion was performed 3 months after the initial surgery from T10 to the pelvis with cement augmentation after removal of the loosened L3-L5 implants. Intra-operative findings were friable tissue and osteoporotic bone. Histology still showed granulomatous inflammation but other cultures were negative. Due to the persistent infection with negative cultures, serology for C burnetii was performed and revealed increased phase I and phase II polyvalent antibodies with titre results of 3200 for both. The patient was diagnosed with chronic Q fever and was given lifelong hydroxychloroquine sulphate 200 mg every 8 hours and doxycycline hyclate 100 mg every 12 hours owing to the presence of the endovascular stent and spinal instrumentation. Serial monitoring of Q fever serology was performed every 6 months. At 2 years after surgery, he remains symptom free with no implant loosening.

First described in 1937, Q fever is caused by C burnetii and can be found worldwide with an overall prevalence of 10%.1 2 Inoculation is through direct contact, ingestion or inhalation of contaminated materials. Traditionally it is thought that contact with cows, goats, sheep or cats causes this disease, but it is not always the case. In chronic Q fever, endocarditis is the most common presentation, seen in 60% to 70% of cases. Other manifestations include hepatitis, pericarditis, myocarditis, vascular infections, and osteoarticular infection.3 Diagnosis is usually by serological testing but can also be on smears or frozen tissue with Giemsa staining that reveals doughnut granulomas. Although serology shows only indirect evidence of infection, it is a simpler and reliable technique. In acute Q fever, testing is performed for antibodies against phase II antigens. In chronic Q fever, testing is for antibodies against phase I antigens. Diagnosis is confirmed if immunoglobulin G titre exceeds 1/800. Cultures are not commonly performed due to its high infectivity and is not available in our laboratory.

Osteoarticular Q fever infections are rare. One study from France showed that only 7.3% of all their patients with Q fever manifested with osteoarticular infection.4 The most common form of Q fever osteoarticular infection is chronic osteomyelitis. This is usually a result of contamination from a previous open fracture or prosthetic joints and vascular grafts.4 Adults usually present with an infected prosthetic joint and children with multifocal osteomyelitis.1

A review of this case revealed that the patient had previously travelled to Guangdong province in China and had contact with farm animals. This was the only potential source of infection from his history. A high index of suspicion is needed for diagnosis, as seen in this case, because a delay in treatment can lead to multiple surgeries. Despite being in an endemic area for spinal tuberculosis,5 other rarer causes of spondylodiscitis with granulomatous inflammation must be considered. These include other bacteria such as Brucellosis, melioidosis, actinomycosis, and Bartonella infections. Spirochetes, fungi, toxoplasmosis, and viruses such as infectious mononucleosis, cytomegalovirus, measles, and mumps are also potential causes. The presence of endovascular infection with spondylodiscitis with granulomatous inflammation and negative cultures should raise the alarm for potential Q fever. Patients are commonly misdiagnosed and treated with prolonged antimicrobials and surgery without improvement.

Concept or design: JPY Cheung.
Acquisition of data: All authors.
Analysis or interpretation of data: All authors.
Drafting of the manuscript: All authors.
Critical revision of the manuscript for important intellectual content: JPY Cheung.

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, JPY Cheung was not involved in the peer review process. Other authors have disclosed no conflicts of interest.

This case report 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 all treatments and procedures and consent for publication.

1. El-Mahallawy HS, Lu G, Kelly P, et al. Q fever in China: a systematic review, 1989-2013. Epidemiol Infect 2015;143:673-81. Crossref

2. Fantoni M, Trecarichi EM, Rossi B, et al. Epidemiological and clinical features of pyogenic spondylodiscitis. Eur Rev Med Pharmacol Sci 2012;16 Suppl 2:2-7.

3. Landais C, Fenollar F, Constantin A, et al. Q fever osteoarticular infection: four new cases and a review of the literature. Eur J Clin Microbiol Infect Dis 2007;26:341-7. Crossref

4. Melenotte C, Protopopescu C, Million M, et al. Clinical features and complications of Coxiella burnetii infections from the French National Reference Center for Q fever. JAMA Netw Open 2018;1:e181580. Crossref

5. Chan-Yeung M, Noertjojo K, Tan J, Chan SL, Tam CM. Tuberculosis in the elderly in Hong Kong. Int J Tuberc Lung Dis 2002;6:771-9.

In June 2020, a 12-year-old boy presented to the emergency department with a 1-day history of retained electrical wire in the urethra. The electrical wire had been self-inserted into the urethra in an attempt to relieve “urethral itchiness”. He reported pain and gross haematuria as a result of this insertion. He and his mother had been unable to remove the wire. Upon admission, physical examination and plain abdominal radiograph revealed a tangle of electrical wire at the penile urethra with 20 cm of wire protruding externally via the urethral meatus (Fig 1a). Removal of the foreign body with 2% xylocaine gel was attempted but failed so emergency surgery was arranged. Despite general anaesthesia, the tangled electrical wire could not be removed by applying traction to the external end. Surgical removal through urethrotomy at the mid-penile urethra was performed (Fig 1b). The wire was fragmented and retrieved completely, and the penile urethra was reconstructed with urethroplasty (Fig 1c).

Upon further discussion with the patient’s mother, she revealed that the patient had been having difficulty settling into school and adapting to everyday life following their recent immigration from Taiwan. With school suspended during the coronavirus disease 2019 (COVID-19) pandemic, the patient was often left alone at home, worsening his emotional health. His mother volunteered that he had no history of insertion of foreign body.

He was assessed by the clinical psychologist who diagnosed limited problem-solving skills and autistic features with a strong interest in using electrical wire. Additional parental support was advised and regular follow-up with the clinical psychologist was arranged. The patient was discharged 2 days after his operation with an indwelling urinary catheter in place for 2 weeks.

In May 2020, a 15-year-old boy was admitted with a 1-day history of a retained plastic bottle in his rectum. He reported recent onset of constipation and had inserted a plastic shampoo container per rectum in the hope of inducing a bowel motion. The patient claimed that the idea for this “regimen” was derived from an internet search for a remedy for constipation while school was suspended due to the COVID-19 pandemic. He had no abdominal pain, per rectal bleeding or fever. Physical examination revealed a soft non-distended abdomen. No mass or foreign body was initially felt per rectum. Plain abdominal radiograph revealed the contour of the plastic shampoo bottle in the region of the transverse colon (Fig 2). He was observed overnight in our surgical ward and repeated per rectal examination the next morning found that the plastic bottle was just palpable with a fingertip. The patient subsequently had a bowel motion with passage of the bottle. He was given advice about diet and the correct use of laxatives for constipation prior to discharge on the same day. His parents were interviewed and advised to pay more attention to their son’s behaviour at home.

Self-insertion of a foreign body into a body orifice is uncommon in children and adolescents.1 Urethral foreign body is even rarer. Motives for teenage intentional foreign body insertion include risk-taking, attention-seeking, sexual gratification, self-injurious behaviour, psychiatric disorder, and self-treatment.2 A wide variety of foreign bodies have been reported in the literature, including needles, safety pins, pencils, ball point pens, wire-like objects (telephone cables, feeding tubes, straws, rubber tubing), toothbrushes, household batteries, and nail scissors.1 In our patients, electrical wire and a plastic bottle were self-inserted in the urethra and rectum, respectively. Patients with urethral foreign body may present with lower urinary tract symptoms including dysuria, haematuria, urethral discharge, and swelling of the external genitalia. Most patients with self-insertion of a foreign body, because of embarrassment, seek medical attention only after the occurrence of complications such as intestinal obstruction, perforation, genital tract infection, or abscess formation.3 Diagnosis can be made in most cases following a comprehensive history taking and physical examination. Radiological evaluation of foreign body with plain radiograph to determine size and number of foreign bodies and their anatomical relationship with surrounding structures usually suffices. Nonetheless ultrasound and computed tomography occasionally may be necessary in selected difficult cases for surgical planning.1 3 Endoscopy (eg, cystoscopy or sigmoidoscopy) is considered the first-line option for confirming and removing the foreign body. Open surgery (eg, laparotomy, urethrotomy, or suprapubic cystotomy) has been described for large foreign bodies or when endoscopic removal is deemed impossible.1 2 3 In our first patient, cystoscopic removal was deemed impossible due to tangling of the electrical wire in the penile urethra; therefore, urethrotomy followed by urethroplasty was necessary to fragment and untangle the wire and achieve complete removal. In addition to removal of the foreign body, this group of teenage patients should be advised to obtain proper psychiatric assessment or psychological support as many of them may have mental health issues.1 2

The COVID-19 pandemic has impacted the mental well-being of children and adolescents as a result of strict public health measures. In Hong Kong, the Education Bureau ordered classes at all kindergartens and primary and secondary schools to close from February 2019 until mid-June. Prevention and Control of Disease Regulation (Cap. 599G) was enforced to restrict the number of persons in group gatherings in public places to maintain social distancing. In mainland China, >20% of college students reported mild to severe anxiety as a result of isolation measures and school suspensions in the midst of the pandemic.4 Lack of contact with peers, reduced opportunities for stress modulation, increased domestic violence, and child maltreatment have been reported as additional threats to the mental health of children and adolescents. These threats have more impact on those already disadvantaged, such as those with physical disabilities, mental health illness, migrant background, or low socio-economic status.5 Our centre observed an increase in foreign body-related conditions during this period, with unusual foreign bodies found at unusual sites. Both patients reported herein were emotionally and socially affected by the COVID-19 pandemic. The mental well-being of children and adolescents should be monitored and supported, and these mental health challenges should be addressed with collaboration among the government, schools, parents, and healthcare professionals.

All authors contributed to the concept or design of the study, acquisition of the data, analysis or interpretation of the 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 case report 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 tenets of the Declaration of Helsinki. Verbal patient consent was obtained.

1. Prasad Ray R, Ghosh B, Pal DK. Urethral foreign body in an adolescent boy: report of two rare cases and review of literature. Int J Adolesc Med Health 2015;27:463-5. Crossref

2. Unruh BT, Nejad SH, Stern TW, Stern TA. Insertion of foreign bodies (polyembolokoilamania): underpinnings and management strategies. Prim Care Companion CNS Disord 2012;14:PCC.11f01192. Crossref

3. Ceran C, Uguralp S. Self-inflicted urethrovesical foreign bodies in children. Case Rep Urol 2012;2012:134358. Crossref

4. Cao W, Fang Z, Hou G, et al. The psychological impact of the COVID-19 epidemic on college students in China. Psychiatry Res 2020;287:112934. Crossref

5. Fegert JM, Vitiello B, Plener PL, Clemens V. Challenges and burden of the Coronavirus 2019 (COVID-19) pandemic for child and adolescent mental health: a narrative review to highlight clinical and research needs in the acute phase and the long return to normality. Child Adolesc Psychiatry Ment Health 2020;14:20. Crossref

A 15-year-old boy with good past health presented with a 1-day history of fever, four episodes of vomiting, and lethargy. He denied diarrhoea, headache, or any neurological symptoms. On arrival, he had blood pressure 106/36 mm Hg, heart rate 127 bpm, temperature 39°C, and oxygen saturation 99% on air. He had reduced pulse volume with normal capillary refilling time. Systemic examination was otherwise unremarkable. Full blood count analysis revealed a haemoglobin level of 14.7 g/dL, white cell count of 9.31 × 10⁹/L, and platelet count of 240 × 10⁹/L. The patient’s blood test results were positive for dengue non-structural protein 1, and negative for dengue immunoglobulin M and immunoglobulin G. His renal profile was normal, with mild raised liver enzymes: alanine aminotransferase 82 U/L and aspartate aminotransferase 310 U/L.

In view of the tachycardia and reduced pulse volume, he was treated as a case of severe dengue fever with compensated shock. He was admitted to the intensive care unit and treated with fluid boluses according to standard protocol. On day 3 of illness he developed severe plasma leakage and was intubated for respiratory distress. His condition soon stabilised and he remained normotensive throughout his stay in intensive care unit. He was extubated on day 7 when he entered a recovery phase. At that time, he had a normal level of consciousness.

Three days later (day 10 of illness), he became encephalopathic, disorientated and able to obey only single step commands. Examination revealed a Glasgow Coma Scale score of E4V4M6 with normal motor power over both upper and lower limbs. A plain computed tomography (CT) brain scan showed hypodensities at bilateral occipital regions and semiovale, predominantly involving the white matter, and suggestive of posterior reversible encephalopathy syndrome (PRES) [Fig 1]. A diagnostic lumbar puncture was offered but refused by his parents. Blood and urine cultures were negative and he was prescribed a 1-week course of intravenous meropenem. He subsequently improved and was discharged well on day 16. Serum dengue polymerase chain reaction was later confirmed as DEN-2. At clinic follow-up, the patient was well and asymptomatic. Repeat CT brain at 3 months showed complete resolution of white matter oedema (Fig 2).

Dengue fever is an important public health problem in the tropics. At present, it is endemic in more than 100 countries. The World Health Organization estimates there to be around 390 million infections per year with 96 million manifesting clinically.1 The clinical features vary from those of a mild febrile illness to severe dengue with systemic complications. Systemic complications include shock, bleeding, plasma leakage, myocarditis and, in some patients, neurological complications. The latter include dengue encephalitis, dengue encephalopathy, meningitis, acute disseminated encephalomyelitis, and Guillain-Barré syndrome.2 Posterior reversible encephalopathy syndrome can occur but is rare.

Posterior reversible encephalopathy syndrome is a distinct clinicoradiological syndrome characterised by headache, seizures, altered consciousness and visual disturbances with the presence of reversible brain vasogenic oedema.3 It can be caused by acute hypertension, hypertensive disease in pregnancy, autoimmune diseases (systemic lupus erythematosus, systemic sclerosis), immunosuppressants, cytotoxic agents, renal failure, and infection. An infectious aetiology such as virus and gram-positive or gram-negative sepsis have all been associated with PRES as has dengue virus.4 5

The pathophysiology of PRES in dengue is still not fully understood. It is postulated to be related to endothelial dysfunction4 that subsequently leads to vasogenic oedema and decreased cerebral blood flow. In addition, inflammatory cytokines can increase vascular permeability with consequent interstitial brain oedema.3 Neuroimaging is essential for diagnosis with characteristics that include vasogenic oedema in the parieto-occipital region of both cerebral hemispheres. The subcortical white matter is always affected. Three primary patterns of magnetic resonance imaging (MRI) brain have been described, namely dominant parieto-occipital pattern, holohemispheric watershed pattern, and superior frontal sulcus pattern. Treatment is mainly supportive with removal of causative factors.

In our patient, diagnosis of PRES was based on clinical and radiological features. He developed acute onset encephalopathy coupled with typical neuroimaging features. Brain CT showed symmetrical hypodensities at bilateral occipital lobes and predominantly involving white matter, suggestive of PRES. He was treated conservatively, and he improved. Follow-up CT brain revealed complete resolution of vasogenic oedema. In terms of aetiology, our patient had no history of hypertension or autoimmune disease or recent cytotoxic agent. In addition, blood pressure remained stable throughout his hospital stay with no decompensated shock. The limitation of our case is the lack of MRI facilities. Compared with CT, MRI better delineates lesions and is preferable if available.

In terms of timing of PRES in relation to dengue fever, PRES has been reported to have occurred on day 5 to 6 of illness in one case,4 and within the non-structural protein 1 antigen positive period (first 7 days of illness) in another.5 In our case, it occurred on day 10 of illness and was considered compatible with the cases reported previously. A diagnosis of PRES secondary to dengue is one of exclusion, supported by typical imaging features and within a reasonable time frame. Alternative causes of PRES should be actively searched for and excluded in every case.

In conclusion, this case highlights PRES as an important differential diagnosis in a dengue patient with neurological deficits. It is important to differentiate PRES from other clinical syndromes since management is mainly supportive and by controlling the underlying conditions. Neuroimaging plays an important role in establishing the diagnosis in the compatible clinical context. The prognosis of PRES is usually good. However, more studies are needed to further elucidate the underlying pathophysiology of PRES in dengue.

Concept or design: SW Cheo and QJ Low.
Acquisition of data: SW Cheo and HJ Wong.
Analysis or interpretation of data: HJ Wong and EK Ng.
Drafting of the manuscript: SW Cheo and QJ Low.
Critical revision of the manuscript for important intellectual content: All authors.

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 disclosed no conflicts of interest.

This case report 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. The patient provided written informed consent for all treatments and procedures, and written consent for publication.

1. World Health Organization. Dengue and severe dengue: Fact sheet. Available from: https://www.who.int/news-room/fact-sheets/detail/dengue-and-severe-dengue.Accessed 4 Nov 2019.

2. Murthy JM. Neurological complications of dengue infection. Neurol India 2010;58:581-4. Crossref

3. Fugate JE, Rabinstein AA. Posterior reversible encephalopathy syndrome: clinical and radiological manifestations, pathophysiology, and outstanding questions. Lancet Neurol 2015;14:914-25. Crossref

4. Mai NT, Phu NH, Nghia HD, et al. Dengue-associated posterior reversible encephalopathy syndrome, Vietnam. Emerg Infect Dis 2018;24:402-4. Crossref

5. Sohoni CA. Bilateral symmetrical parieto occipital involvement in dengue infection. Ann Indian Acad Neurol 2015;18:358-9. Crossref

Ureterosciatic hernia is an extremely rare cause of ureteral obstruction. We report a patient with left pyonephrosis and obstructive uropathy caused by a ureterosciatic hernia.

A 97-year-old woman with a history of ischaemic stroke, hypertension, hyperlipidaemia, and chronic obstructive pulmonary disease attended the Accident and Emergency department of Kwong Wah Hospital in January 2020 with a 1-day history of decreased general health and vomiting. She had no urinary or bowel symptoms. Her vital signs were stable on admission and physical examination of her cardiorespiratory and abdominal systems was unremarkable.

Blood tests on admission revealed acute kidney injury with serum creatinine level of 387 μmol/L (baseline 73 μmol/L), leucocytosis (23.8 × 10⁹/L), and elevated C-reactive protein level of 287 mg/L.

Kidney, ureter, and bladder plain radiograph revealed the absence of urinary stone. Amoxicillin with clavulanic acid were administered intravenously. The following day the patient developed fever up to 40.1°C with abdominal pain. Urgent contrast computed tomography scan of the abdomen and pelvis showed moderate-to-severe left hydronephrosis (Fig 1) with hydroureter up to 1.4 cm down to the distal ureter at the level of the left greater sciatic foramen, where the ureter herniated out of the pelvis with an abrupt calibre change (Fig 2). There was reduced enhancement of the left kidney with no contrast excretion evident on the delayed phase. Retrograde ureteral stenting was performed and 30 mL pus was drained from the left kidney.

Blood and urine cultures grew Escherichia coli. The patient recovered gradually with a 2-week course of antibiotics. The double-J stent was removed at the end of March (2 months after stent insertion). However, she developed another episode of urosepsis shortly afterwards and a double-J stent was re-inserted. She remained well with regular revision of double-J stent.

Ureterosciatic hernia is a rare disease with no more than 40 cases published worldwide to date.1 It occurs when the peritoneal sac and its contents (eg, ovary, small intestine, colon, greater omentum) protrude through the greater or lesser sciatic foramen.2 When the ureter becomes involved in the herniated contents, it is called ureterosciatic hernia. The sacrospinous ligament divides the sciatic notch into the greater and lesser sciatic foramina. The piriformis muscle subdivides the greater sciatic foramen into the suprapiriformis and infrapiriformis compartments. Ureterosciatic herniation commonly occurs through the suprapiriformis compartment of the greater sciatic foramen.2 Herniation of the ureter into the greater sciatic foramen often results from piriformis muscle weakening due to increased pressure in the intra-abdominal area due to pregnancy, constipation, surgery, trauma, neuromuscular diseases, or hip disease.3 It commonly develops in elderly women due to their wider pelvic bones but congenital cases have also been reported. The left ureter is affected more often than the right.4

The clinical presentation of ureterosciatic hernia varies and can range from asymptomatic to life-threatening urinary sepsis or obstructive uropathy. Patients may complain of flank or abdominal pain, nausea, and vomiting. A palpable mass over the affected area may be present.2

Since the signs and symptoms of ureterosciatic hernias are rather non-specific, the diagnosis is commonly made by radiographic studies including intravenous urogram, retrograde pyelography, and computed tomography. Obstruction of the ureter with U-shaped tortuosity into the sciatic foramina, also referred to as a “curlicue ureter” sign, is suggestive of ureterosciatic hernia.5

Treatment options include observation (mostly for asymptomatic patients), ureteral stenting, and surgical correction. Open, laparoscopic and robotic approaches have been described for reduction of the ureter and hernia repair.1 As our patient was aged 97 years with multiple co-morbidities, surgery was not considered.

Although seldom seen, ureterosciatic hernias are occasionally encountered. Diagnosis can be a challenge given its non-specific symptomatology and rarity.

Concept or design: All authors.
Acquisition of data: CYC Chan.
Analysis or interpretation of data: CYC Chan.
Drafting of the manuscript: CYC Chan and TCT Lai.
Critical revision of the manuscript for important intellectual content: CYC Chan.

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 case report 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. The procedure was done under 2MO consent as the patient’s condition was unstable at the time. Risks of and indications for the procedure were discussed with the patient’s daughter who showed understanding and agreed to proceed. The patient provided consent for publication.

1. Kamisawa K, Ohigashi T, Omura M, Takamatsu K, Matsui Z. Ureterosciatic hernia treated with laparoscopic intraperitonization of the ureter. J Endourol Case Rep 2020;6:150-2. Crossref

2. Gandhi J, Lee MY, Joshi G, Smith NL, Khan SA. Ureterosciatic hernia: an up-to-date overview of evaluation and management. Translational Research in Anatomy 2018;11:5-9. Crossref

3. Gee J, Munson JL, Smith JJ 3rd. Laparoscopic repair of ureterosciatic hernia. Urology 1999;54:730-3. Crossref

4. Loffroy R, Bry J, Guiu B, et al. Ureterosciatic hernia: a rare cause of ureteral obstruction visualized by multislice helical computed tomography. Urology 2007;69:385.e1-3. Crossref

5. Pollack HM, Popky GL, Blumberg ML. Hernias of the ureter—an anatomic-roentgenographic study. Radiology 1975;117:275-81. Crossref