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

In December 2016, a 47-year-old Chinese woman was referred to our clinic for treatment of a large cystic pancreatic lesion. She presented with a 12-month history of intermittent epigastric pain associated with eating and weight loss of 5 kg. The patient had a history of uterine fibroids and underwent myomectomy 10 years previously. She was also under observation for a benign thyroid nodule. She had no family history of malignancy, denied consumption of alcohol, was a non-smoker, and lived as a housewife.

A contrast-enhanced computed tomography scan of the abdomen revealed a cystic neoplasm 8 cm in diameter involving the head, neck, and body of the pancreas, with proximal dilatation of the pancreatic duct (Fig 1). Based on the patient’s symptoms and the size of the lesion, surgical resection was recommended. Preoperative laboratory test results, including full blood count, liver and renal function tests, blood glucose level, amylase, carcinoembryonic antigen, and cancer antigen 19.9, were within normal limits.

During surgery, a multiloculated cystic lesion was identified (Fig 2). The cystic content was serous. No nearby enlarged lymph nodes or tissue invasion suggestive of frank malignancy were evident. The pancreatic tail was healthy. An extended middle pancreatectomy (EMP) was performed. The patient recovered without complication and was discharged from hospital 13 days after surgery. Histological examination of the resected tissue confirmed a benign serous cystadenoma.

A bilateral subcostal incision was made. The lesser sac was entered to expose the anterior aspect of the pancreas after division of the gastrocolic ligament (Fig 2a). The gastroduodenal junction was purposefully divided to gain better exposure of the large cystic lesion and to facilitate further pancreatic dissection (Fig 2b). The posterior peritoneum along the inferior and superior margins of the pancreas was dissected. The superior mesenteric vein was identified under the neck of the pancreas. The splenic vein was carefully divided away from the gland and all the small branches of the pancreas draining into the splenic vein were ligated. The gallbladder was removed to facilitate identification and mobilisation of the common bile duct, the hepatic artery, and the portal vein from above. The involved portion of the pancreas was mobilised on both cephalic and caudal sides. The cystic tumour was resected in its entirety with a margin by scalpel and cautery. Most of the head (estimated 80%-90%), neck, and body of the pancreas were removed (Fig 2c). The remaining pancreatic head stump was carefully over-sewn with 3-O Prolene sutures (Ethicon; Cornelia [GA], United States) to avoid pancreatic leak. The pancreatic duct opening was identified and separately sutured. The stump was further re-enforced with fibrin sealant, Tisseel glue (Baxter Healthcare, Deerfield [IL], United States). The distal side stump was anastomosed into a Roux loop of jejunum using an end-to-end technique with invagination (Fig 2d). A short segment of catheter was inserted into the pancreatic duct. The gastric pylorus was then joined up to the proximal jejunum as a gastrojejunostomy and a jejunojejunostomy was fashioned to prevent bile reflux. The proximal jejunum was reconnected to the jejunal Roux loop distally. Two drainage tubes were placed close to the closed cephalic stump and the pancreaticojejunostomy anastomosis before proceeding with abdominal wound closure.

For benign or low-grade malignant lesions of the pancreatic neck and body, traditional approaches for surgical resection include a pancreaticoduodenectomy (PD) or an extended distal pancreatectomy. Enucleation is suitable only for small and superficial lesions that do not have any connection with the pancreatic duct. In 1957, Guillemin and Bessot first reported the technique of middle pancreatectomy (MP) for chronic pancreatitis and pancreatic transection injury.1 In 1982, Dagradi and Serio proposed the use of MP for resection of benign tumours or tumours with low malignant potential situated in the pancreatic neck and body.2 The technique has since gained acceptance.

In this case, the large cystadenoma involved a significant portion of the head of the pancreas in addition to the neck and the body. Traditional approaches for resection of such a lesion would involve either subtotal pancreatectomy or PD. The MP technique described in the literature has not included resection of the head of the pancreas. However, in this case, most of the pancreatic head in addition to the neck and the body of pancreas had to be resected. Therefore, the procedure is being formally named for the first time, EMP.

Extended middle pancreatectomy permits resection of a lesion that extends into the head of the pancreas while conferring similar advantages of an MP over traditional approaches. Compared with other surgical options (subtotal pancreatectomy and PD), this confers the advantage of parenchymal preservation and consequent preservation of pancreatic exocrine and endocrine functions.3 Pancreaticoduodenectomy has been associated with increased postoperative morbidity and mortality. Similarly, the inability to preserve the spleen in subtotal pancreatectomy has been associated with complications of thrombosis and susceptibility to infection. Finally, MP has been associated with a higher incidence of pancreatic fistula, compared with PD and extended distal pancreatectomy procedures.4 5 This has been attributed to the need to manage two pancreatic remnants by anastomosis or closure. However, as much less pancreatic head tissue is left behind in EMP, the risk of pancreatic leak from the proximal stump should theoretically be lower than that observed in MP.

We report, to the best of our knowledge, the first clinical case of EMP in the management of a large cystic neoplasm. This case demonstrated excellent postoperative outcomes and suggests that EMP may be considered a viable preferred surgical option in selected cases. However, the technical difficulties of this procedure should not be underestimated. The long-term functional outcomes have yet to be substantiated by further clinical experience.

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 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, AKK Chui was not involved in the peer review process. The 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 tenets of the Declaration of Helsinki. Patient consent was obtained.

1. Guillemin P, Bessot M. Chronic calcifying pancreatitis in renal tuberculosis: pancreatojejunostomy using an original technic [in French]. Mem Acad Chir (Paris) 1957;83:869-71.

2. Dagradi A, Serio G. Pancreatectomia intermedia. In: Enciclopedia Medica Italiana. Pancreas, vol XI. Florence: USES Ed Scientifiche; 1984: 850-1.

3. Tan Z, Chen P, Dong Z, Zhou B, Guo WD. Clinical efficacy of middle pancreatectomy contrasts distal pancreatectomy: a single-institution experience and review of literature. ANZ J Surg 2019;89:E184-9. Crossref

4. Shibata S, Sato T, Andoh H, et al. Outcomes and indications of segmental pancreatectomy. Comparison with distal pancreatectomy. Dig Surg 2004;21:48-53. Crossref

5. Du ZY, Chen S, Han BS, Shen BY, Liu YB, Peng CH. Middle segmental pancreatectomy: a safe and organ-preserving option foe benign and low-grade malignant lesions. World J Gastroenterol 2013;19:1458-65. Crossref

A 62-year-old man with no known systemic disease was brought to an emergency department by ambulance. He was found trapped in a car after colliding with a bridge and was found to have been driving under the influence of alcohol. On arrival, he complained of headache, neck pain, and chest pain. Physical examination revealed blood pressure of 133/71 mm Hg, heart rate 71 beats per minute, respiratory rate 22 breaths per minute, and oxygen saturation on air of 93%. He appeared intoxicated and was disorientated with a score of 13 (E3V4M6) on the Glasgow Coma Scale. His pupil size was 3.5 mm with bilateral normal reaction to light. A thorough neurological examination could not be performed but the patient was able to move all four limbs without limitation. Nonetheless a scalp laceration, upper back tenderness and left lower cervical crepitus were evident during palpation. Chest examination revealed bilateral equal breath sounds but some basal crackles and a left flank abrasion about 3 × 4 cm in size. Haemogram and biochemistry test results were within normal limits but serum alcohol level was elevated at 2.48 g/L. Chest radiography revealed a posterior fracture at the left fourth to sixth ribs and right ninth rib as well as left cervical subcutaneous emphysema that raised the suspicion of barotrauma associated with chest trauma (Fig 1). In view of the possibility of barotrauma and intracranial injuries, further evaluation with computed tomography (CT) of the brain and chest was arranged. The former was normal and the latter confirmed right first to second posterior ribs and left third to sixth posterior ribs fractures, as well as a small left-sided pneumothorax, bilateral pulmonary contusions, and pneumomediastinum. Notably, free air accumulated in the posterior epidural spaces of the cervical and thoracic spine as well as in the left retrospinal and paraspinal muscle layers with accompanying left second and third thoracic spine posterior neural arch fracture in cervical spine CT scan (Fig 2); these findings were compatible with epidural pneumorrhachis. Additionally, air was seen dissecting from the posterior neck into the epidural space. A neurosurgeon recommended close observation rather than surgical intervention for both the epidural pneumorrhachis and thoracic spine fracture. The patient was admitted to the thoracic surgical ward where he received conservative therapy for rib fractures, pneumothorax and pneumomediastinum including meperidine for pain control and oxygen therapy via a nasal cannula at 3 L/min. New chest radiographs were taken on day 4 after surgery. Cervical emphysema had mostly resolved but there was progression of pulmonary contusion with bilateral minimal pleural effusions. The patient also exhibited wheezing and dyspnoea that warranted inhaled bronchodilators and systemic steroid therapy. The patient was discharged on day 12 after surgery with symptom improvement; no neurological sequelae were noted during follow-up at the out-patient department.

Pneumorrachis (PRS) is characterised by the presence of air within the spinal canal and can be classified according to its location as subarachnoid or epidural PRS. Each is associated with different pathophysiological mechanisms and causes. It may result non-traumatically from spinal degeneration, gas-producing infections, or spontaneous pneumomediastinum in patients with asthma. Iatrogenic PRS may occur following a surgical procedure, epidural anaesthesia, or lumbar puncture but PRS following trauma is rare.1 Almost all types of PRS are found in association with air distribution in other compartments and cavities of the body. For example, PRS is observed in conjunction with pneumocephalus, pneumothorax, pneumomediastinum, pneumopericardium, or subcutaneous emphysema. Pneumorrachis following trauma is rare and arises from the presence of air in the posterior mediastinum or retropharyngeal space dissecting along the fascial planes from the posterior mediastinum or retropharyngeal space through the neural foramina into the epidural space under the driving pressure of a pneumothorax and pneumomediastinum and low resistance of loose connective tissue.2

Epidural PRS has been reported to occur most commonly secondary to traumatic pneumomediastinum. Neurological deficits have not been reported in epidural PRS secondary to trauma. In contrast, traumatic subarachnoid PRS is almost always secondary to major trauma and skull bone fractures, and indicates severe injury and possible association with complications such as tension pneumocephalus or meningitis.3 Various neurological deficits, such as acute lumbar root compression, chronic radiculopathy, and cauda equina syndrome may occur. It is therefore important to identify air in the epidural space and subarachnoid space to enable prompt and adequate management.4 The possible mechanism of PRS in our patient was air entry into the epidural space because of pneumomediastinum, left-sided pneumothorax and subcutaneous emphysema extending through the fractured neural arch of the thoracic spines into the epidural space; these conditions fulfil the criteria for benign traumatic PRS. Criteria for a diagnosis of benign traumatic epidural PRS include substantial pneumomediastinum with extensive subcutaneous emphysema due to thoracic trauma.4 There may also be incidental findings of small amounts of epidural air demonstrable only on CT and unrelated to spinal trauma. “Benign” epidural PRS can be managed conservatively with spontaneous resolution of epidural air.4 Repeat or further neuroimaging studies are not routinely provided in cases of epidural PRS, provided there is no new or progressive neurological deficit. Three cases have been reported with complete resolution of PRS on follow-up CT scan after 4 to 14 days.5 6 7

The PRS is usually asymptomatic and an accidental finding on CT images while evaluating other severe or life-threatening torso injuries. Although CT is the gold standard for diagnosis, it is difficult to differentiate epidural and subarachnoid PRS. Nonetheless in the former, air is collected at the corner or peripherally unlike subarachnoid PRS where it is distributed more centrally in the canal based on normal anatomy. Magnetic resonance imaging or intrathecal contrast CT can distinguish intradural from extradural air.7 In most cases of epidural PRS, the air reabsorbs spontaneously without recurrence and the patient can be managed conservatively.1 In subarachnoid PRS, timely consultation with a neurosurgeon for decompression of nerve root injuries, tension pneumocephalus and exploratory surgery by a chest surgeon to detect severe thoracic trauma is vital. Impressively, Avci et al5 reported a case of complete resolution of subarachnoid PRS on the fifth day of admission with recovery of neurological deficit after chest tube insertion. They concluded that the negative pressure of the chest drain to withdraw the air supported the hypothesis of a one-way valve mechanism in which high pressure air travels through fascial layers but is unable to return. The routine use of antibiotic is not recommended in either type unless meningitis develops.1

Traumatic PRS is rare and an incidental radiological finding. Epidural PRS is often self-limiting with a good prognosis. Although most patients can be managed conservatively, prompt recognition of subarachnoid PRS with neurological deficits and other concurrent life-threatening torso injuries is vital.

Concept or design: KH Lee.
Acquisition of data: YY Yang.
Analysis or interpretation of data: CB Chua.
Drafting of the manuscript: YY Yang and KH Lee.
Critical revision of the manuscript for important intellectual content: CW Hsu.

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.

We would like to extend our special thanks to Dr Cheng-yang Lee, Department of Medical Imaging, Chi Mei Medical Center, Tainan, Taiwan who provided insight and expertise on the image interpretation that greatly improved our manuscript.

All authors have no commercial association, such as consultancies, stock ownership or other equity interests or patent-licensing arrangements.

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. Oertel MF, Korinth MC, Reinges MH, Krings T, Terbeck S, Gilsbach JM. Pathogenesis, diagnosis and management of pneumorrhachis. Eur Spine J 2006;15 Suppl 5:636-43. Crossref

2. Defouilloy C, Galy C, Lobjoie E, Strunski V, Ossart M. Epidurual pneumatosis: A benign complication of benign pneumomediastinum. Eur Respir J 1995;8:1806-7. Crossref

3. Goh BK, Yeo AW. Traumatic pneumorrhachis. J Trauma 2005;58:875-9. Crossref

4. Willing SJ. Epidural pneumatosis: a benign entity in trauma patients. AJNR Am J Neuroradiol 1991;12:345.

5. Avci İ, Başkurt O, Şirinoğlu D, Aydin MV. Rapid disappearance of pneumorrhachis after chest tube placement. Turk J Emerg Med 2019;19:146-8. Crossref

6. Kim SW, Seo HJ. Symptomatic epidural pneumorrhachis: a rare entity. J Korean Neurosurg Soc 2013;54:65-7. Crossref

7. Pfeifle C, Henkelmann R, von der Höh N, et al. Traumatic pneumorrhachis. Injury 2020;51:267-70. Crossref