Gastric outlet obstruction (GOO) is unusual and must be suspected in children with chronic vomiting and abnormal weight status. The treatment depends on etiology, and surgery is not always the first remedy. Diagnosis is easily confirmed by upper gastrointestinal fibroscopy.We report the case of an 11-year-old girl, who was presented with non-bilious emesis and weight loss. Abdominal computed tomography, ultrasound, and upper gastrointestinal fibroscopy showed dilated stomach with pyloric stenosis, which was confirmed by abdominal laparoscopic exploration and cured by Heineke-Mikulicz pyloroplasty. We also compare our study to previously reported cases. Gastric outlet obstruction (GOO) is rare in children, except in cases due to infantile hypertrophic pyloric stenosis. Primary acquired GOO has been seldom reported and consequently remains poorly understood. Moore has classified congenital obstructions in 1986, including antral and pyloric atresia and webs . We descHelicobacter pylori infection was negative. Upper GI revealed an important gastric stasis and an incomplete pyloro-duodenal stenosis with a normal duodeno-jejuno-ileal flow . The abdominal examination was normal, and there were no other coexisting problems. A biological checkup revealed anemia (hemoglobin: 8.8 g/dL) and hypoprotidemia (53 g/L). Abdominal computed tomography was performed, which showed a significant gastric stasis upstream a duodenal stenosis  . EpidermIf the peptic origin cannot be clearly ruled out, other etiologies have to be considered. Congenital disease is reported in about 1 per 100,000 newborns, including pyloric atresia, and pyloric and antric webs. In case of complete obstruction, antenatal diagnosis is possible, and abdominal ultrasound shows a voluminous stomach and hydramnios. The symptoms appear during the neonatal period, with non-bilious emesis . ConcernSurgery is rare in pyloric stenosis, in which the etiology is peptic, and can be proposed after several weeks of medical treatment. However, when the stenosis is congenital and tardily revealed, or acquired GOO is idiopathic, the surgery has to be suggested straight away. Heineke-Mikulicz pyloroplasty remains the standard curative treatment in most reported cases, which was the main treatment for our patient, and this procedure can now be performed laparoscopically .In 2006, Hameury et al. found that the pneumatic dilatation was ineffective and that the cure was completed by surgery . In 2007We compare our study to a similar case reported by Härter et al. in 2017 of a five-year-old boy with non-bilious vomiting and a slightly reduced nutritional status . In the Other causes of GOO are classified by Moore as follows: antral gap atresia, pyloric gap atresia, pyloric septum, pyloric membrane, antral membrane, or web . TreatmePediatric GOOs are uncommon. Their causes and treatments are debated. Non-bilious vomiting with weight loss must be explored in children out of the usual age of hypertrophic pyloric stenosis. Fibroscopy and upper gastrointestinal are essential to approve the diagnosis. In case of medical treatment failure, the pyloroplasty allows most often to solve the problem.


Sinonasal malignant melanoma (SMM) is a rare malignant tumour among head and neck cancers predominantly found in adults 60 years and above. The commonly reported symptoms for sinonasal tumour lesions are nasal obstruction and recurrent, painless epistaxis as the symptoms are non-specific and can delay the diagnosis. Moreover, melanoma has a poor prognosis regardless of its location. We report an 86-year-old female patient presenting with recurrent, painless epistaxis from the nasal cavity. Anterior rhinoscopic examination revealed a bluish-black, bleeding mass completely obstructing the left nasal nare. Contrast-enhanced computed tomography of the nasal cavity and sinus region showed a polypoidal soft tissue attenuation with heterogeneous enhancement completely filling the left nasal cavity. The patient underwent endoscopic excision. Histopathology of the specimen showed a small, round and blue cell tumour which immunohistochemistry found to be positive for S100 and HMB 45. After surgical resection, the patient received chemotherapy and radiotherapy. Sinonasal malignant melanoma is a rare, aggressive tumour that has a very poor prognosis. Contrast-enhanced computed tomography of the nasal cavity and paranasal sinuses is the imaging modality of choice which reveals the enhancing mass. There is no optimal management strategy for SMM. Surgical resection is the first-line treatment but is limited due to the complex anatomy of the sinonasal region. Melanocytes are neural crest cell derivatives that are found all over cutaneous and mucosal surfaces . In the An 86-year-old female presented to the otorhinolaryngology clinic in August 2021, with a chief complaint of recurrent episodes of bleeding from both nostrils for the past two weeks. The bleeding was not active at the time of presentation. The patient gave a history of hypertension and dyslipidemia. The hypertension was diagnosed eight years ago and she was prescribed perindopril and atenolol while the dyslipidemia was diagnosed four years ago for which she was prescribed atorvastatin. There was no history of trauma or inserted foreign bodies. Anterior rhinoscopy examination revealed a bluish black mass occupying the left nasal cavity and minimal ulcerative changes on the left lateral dorsum of the nose in contrast to the right nasal cavity which was normal on examination. Examination of the ears revealed normal bilateral tympanic membranes and no signs of hearing impairment. In addition, general physical examination was insignificant, and the vital signs were within normal range.Contrast-enhanced computed tomography (CT) of the nasal cavity and paranasal sinuses showed a heterogenous-enhancing left nasal cavity mass, involving the turbinate as well as obliteration of osteomata complex and extension into posterior nasopharynx causing a mass effect on the medial wall of the maxillary sinus, leading to obstruction associated with diffuse mucosal thickening of the left maxillary sinus with extension superiorly to the ethmoid sinuses. Mucosal thickening was also evident in both ethmoid sinuses and the left frontal sinus Figure . The lamThe patient underwent an endoscopic resection under general anaesthesia. Intra-operative findings showed a friable, sessile mass arising from the left lateral wall of the nose. Haemostasis was achieved. The surgery was uneventful and the patient was discharged after two days of hospitalisation. Histopathology of the specimen revealed a malignant tumour composed of sheets of round to oval tumour cells with spindle, pleomorphic, rhabdoid, plasmacytoid and small cell cytoplasm and regular round hyperchromatic nuclei, melanin pigment was seen at places suggestive of small round blue cell tumour which can be considered a variant of sinonasal melanoma. Immunohistochemistry of the biopsy was found to be positive for S100 and HMB 45 Figures -3. AfterMelanoma of the head and neck are uncommon, accounting for 0.4% to 1.8% of all malignant melanoma. The most common sites for melanomas are the anterior portion of the nasal septum, followed by the lateral wall of the nasal cavity . SinonasTo evaluate staging, location, and regional extension as well as to rule out lesions affecting the meninges, brain tissue, and major arterial structures, CT and magnetic resonance imaging are required. Displacement of the periorbita is typically diagnosed on CT, with a reported negative predictive value (NPV) of 86% and a positive predictive value (PPV) of 75% . ComputeTreatment of recurrent epistaxis of unknown origin with packing/cautery should be followed by timely endoscopy with potential biopsy and then imaging. The prognosis and available treatments for sinonasal melanoma are significantly impacted by early diagnosis. Imaging latency could delay diagnosis and therapy and have a negative impact on the course of the disease. We advise primary care doctors to promptly refer patients who experience frequent bouts of epistaxis for additional attention or to order imaging exams for their sinuses.Sinonasal malignant melanoma is a rare, aggressive tumour that has a very poor prognosis. Contrast-enhanced computed tomography of the nasal cavity and paranasal sinuses is the imaging modality of choice which reveals an enhancing mass. Surgical resection is the first-line treatment which has its own limitations due to limited surgical visibility and the complex anatomy of the sinonasal region. Surgery in combination with radiotherapy and adjuvant chemotherapy is utilised, however, it does not always improve chances of survival as local recurrence and distant metastasis are common outcomes and are poor prognostic indicators of the disease.


Glioblastoma (GBM) is difficult to cure with conventional multimodal treatment and has an extremely poor prognosis. Boron neutron capture therapy (BNCT) is a new particle therapy for malignant tumors in the brain and head and neck region. This radiotherapy utilizes a nuclear reaction between neutrons and a nonradioactive isotope, boron-10. In this method, a boron compound is administered transvenously into the body. The boron compound has the property of being selectively taken up only by the cells of malignant tumors, and the subsequent irradiation with neutrons can destroy malignant tumor cells without damaging normal cells. Since the irradiation dose to normal tissues is reduced in BNCT, it may be possible to re-irradiate malignant tumors that recur after radiotherapy. Clinical trials have reported prolonged survival and safety of BNCT in a small number of patients with refractory malignancies, including GBM, but these reports do not address quality of life or activities of daily living (ADL) after treatment, and there is no information on the assessment of local control by imaging. Here, we report a case of GBM that recurred after surgery, 60 Gy of conventional radiotherapy and standard treatment with temozolomide. The patient achieved long-term local control and survival over five years after BNCT and was able to maintain ADL at home without any specialist care. We describe the case with evaluation using longitudinal magnetic resonance imaging (MRI). Glioblastoma multiforme (GBM) accounts for approximately 25-40% of central nervous system malignant tumors and is the most common primary malignant brain tumor ,2. This Boron neutron capture therapy (BNCT) is a new particle therapy for malignant tumors in the brain, head and neck regions. This radiotherapy utilizes a nuclear reaction between neutrons and a nonradioactive isotope, boron isotope (10B). In this method, a boron compound is administered transvenously into the body. The boron compound has the property of being selectively taken up only by the cells of malignant tumors, and the subsequent irradiation with neutrons can destroy malignant tumor cells without damaging normal cells. Since the total irradiation dose to normal tissues is reduced in BNCT, it may be possible to re-irradiate malignant tumors that recur after radiotherapy. Clinical studies have reported prolonged survival and safety of BNCT in a small number of patients with refractory malignancies, including GBM ,9. Howev6-methylguanine-DNA methyltransferase (MGMT) were not performed. Postoperatively, the patient received radiotherapy of 60 Gy/30 fractions and temozolomide (140 mg) for 42 days. The patient remained mildly aphasic but was able to communicate well and maintain ADLs at home. However, three months after surgery, local regrowth of the tumor was observed and recurrence was diagnosed. The patient was referred to our hospital and underwent BNCT with the locally recurrent tumor as the target volume. The patient had a fever for three days after irradiation as an acute adverse event, which resolved spontaneously. Although he was able to live at home without any medical or nursing assistance after the completion of the first surgery, one month after the BNCT, he was hospitalized for the need of assistance with walking. Subsequently, the patient suffered brain necrosis, for which surgical and medical intervention was required. Pathological diagnosis of the resected specimen revealed radiation necrosis. Bevacizumab was used for radiation brain necrosis. Four years and six months (54 months) later, the patient was diagnosed with recurrence, and after five years and two months (62 months), his general condition deteriorated due to the primary disease. Survival for this patient from the initial diagnosis was five years and eight months (68 months). The patient was able to live at home until death.A 59-year-old man was diagnosed with a brain tumor in the left temporal lobe by head CT and MRI after the development of a speech disorder. The tumor was removed by craniotomy. The pathological diagnosis was GBM WHO grade IV. At the time, more than 10 years ago, genetic mutation tests for isocitrate dehydrogenase (IDH) and OBNCT procedureBNCT is an advanced type of radiotherapy, in which the radiation beam consists of two heavy particle beams produced by a reaction of a stable boron isotope 10B) with low-energy thermal neutrons. The reaction process is illustrated in Figure 0B with lBNCT enables irradiation of tumor cells with thermal neutrons extracted from a nuclear reactor and is used clinically primarily for locally recurrent tumors after radiotherapy. In clinical practice, external irradiation of slightly higher energy epithermal neutrons is used to improve the depth of field. These epithermal neutrons repeatedly collide elastically with hydrogen nuclei in the body, losing energy and becoming thermal neutrons which react with 10B absorbed by tumor cells. In this case, the dose distribution during BNCT is shown in Figure Boronophenylalanine (BPA) is often used as a boron compound in BNCT. In this study, PET (Positron Emission Tomography) diagnosis with 18F-fluoro-BPA (18F-BPA) was performed before BNCT to confirm the accumulation of BPA in tumor tissue, and BNCT was indicated when the accumulation was about three times greater than that in the surrounding normal brain. In this case, the dose was calculated in advance by simulating the maximum dose in the normal brain, as shown in Table Imaging evaluationDuring the follow-up, the MRI scan was performed every 3-4 months, starting just before BNCT. The image immediately before BNCT showed a contrast-enhanced lesion at the surgical wound margin in the left temporal lobe in Figure In contrast-enhanced MRI at one month after BNCT, the contrast-enhancement effect for the surgical wound appeared to be slightly reduced. And it was difficult to determine if the tumor was enlarged Figure . At threContrast-enhanced MRI at 24 months after BNCT showed a further weakening of the limbic contrast effect and improvement of edema and swelling in the cerebral hemispheres, with no evidence of tumor recurrence Figure . At 30 mContrast-enhanced MRI at 54 months after BNCT Figure  showed tSurgical resection is the most effective treatment for benign tumors and low-grade gliomas, but GBMs are difficult to resect because of their invasive growth . In addiThe present patient had a relatively large tumor and was classified as intermediate-risk . Thus, tIn the use of BNCT for brain tumors, Kageji et al. reported a median survival time (MST) of 19.5 months for 23 patients with first-episode glioblastoma treated with BNCT alone . The resThis case suggests that tumor control and maintenance of ADLs in long-term survival after recurrent GBM are possible with BNCT if adverse events of radiation dermatitis and brain necrosis can be overcome.


Retroperitoneal sarcomas represent a group of rare malignant neoplasms with complex clinical management and often a poor prognosis. An elderly male presented with a slowly progressive, right-sided abdominal lump for four months associated with loss of appetite and abdominal discomfort. Abdominal examination revealed an apparent retroperitoneal lump in the right lumbar and umbilical region, which was well-defined, and firm in consistency with the bosselated surface. Contrast-enhanced computed tomography (CECT) of the abdomen and pelvis revealed a heterogenous lobulated malignant appearing retroperitoneal lesion arising from the right anterior perirenal space with a differential of retroperitoneal sarcoma. Wide local excision of the tumor was done. Histopathology of the lesion revealed a smooth muscle tumor of uncertain malignant potential (STUMP). The patient is asymptomatic and recurrence-free after 24 months of follow-up. Retroperitoneal sarcomas represent a group of rare malignant neoplasms, complex clinical management, and often a poor prognosis with about 70 different histological variants and an annual incidence of 0.76 new cases per 100,000 . Rare reA well-built gentleman in his 60s, without any comorbidity, presented to the general surgery outpatient department with a painless, non-progressive lump in the right side of the abdomen, noticed four months ago, without a history of fever, vomiting, and abdominal distension. On examination, the size of the lump was 11 x 15 cm in the right lumbar and umbilical region, located in the retroperitoneum, which was apparent on inspection, with ill-defined margins, firm consistency, and bosselated surface on palpation. General physical examination was unremarkable with no inguinal or cervical lymphadenopathy. Ultrasonography (USG) of the abdomen and pelvis was done as an adjunct to clinical examination suggesting a heteroechoic mass of size 10 x 15 cm, in the retroperitoneum. Contrast-enhanced computed tomography (CECT) of the abdomen revealed a well-defined, lobulated heterogeneous lesion of size 11.6 x 11.2 x 15 cm in the retroperitoneum and right anterior perirenal space. Inferolateraly, the lesion was seen infiltrating the right psoas muscle with focal loss of the fat plane. Laterally, the lesion is seen abutting the inferior vena cava (IVC), right ureter, and adjacent bowel loops without infiltration. No fat or calcification was noted within the lesion  mass. The outer surface of the mass was smooth and well-encapsulated with congested blood vessels (Figure The cut surface was bosselated, nodular, solid homogenous, greyish-white mass with myxoid and cystic areas. The largest cyst measured 5 x 6 x 4 cm, which yields straw-colored fluid. The findings were consistent with the CECT done preoperatively. The postoperative microscopic findings included tumor mass of variable cellularity with spindle cells arranged in a storiform pattern. These spindle cells are large, with a severe pleomorphic nucleus, vesicular chromatin, prominent nucleoli, and a moderate amount of cytoplasm, in an abundant collagenous stroma (Figure Upon vimentin stain, nuclear and cytoplasmic positivity was seen, suggestive of mesenchymal origin Figure .H-caldesmon stain was also positive, narrowing the diagnosis down to smooth muscle tissue. Tumor cells were also diffusely positive for smooth muscle actin and desmin, PanCK, S-100, WT-1, and ALK-negative. There were areas of necrosis corresponding to less than 50% of tumors noted. Mitotic count was 4-5 figures/high power field. Margins were free of tumors. The patient is in regular follow-up at an interval of every six months and was last reviewed after 24 months of surgery, and he was asymptomatic without recurrence which was confirmed after clinical and radiological investigations. The multidisciplinary tumor board discussion suggested no role of adjuvant therapy at present but to keep the patient in close follow-up.Primary retroperitoneal neoplasms are a rare, heterogenous group of tumors not arising from any retroperitoneal organ . These lA retroperitoneal sarcoma is a rare group of soft tissue sarcoma with varied presentation and complex histological categories associated with poor prognosis. Soft tissue tumor of uncertain malignant potential is even rare entity that can neither be categorized as benign nor as a malignant entity. Surgery after multidisciplinary tumor board discussion and strict follow-up adherence can have good promising outcomes.


Paragangliomas are tumors of the autonomic nervous system, arising from paraganglionic tissue. Paragangliomas of the head and neck region are very rare. In the head and neck, the most common sites of origin of this neoplasm are the carotid body, the jugular bulb and the vagal body. Paragangliomas of the nose and paranasal sinuses are very uncommon. The authors referred one case of nasal paraganglioma in a 45-year-old male patient, who was submitted to surgical excision, and included clinical findings, diagnostic criteria, treatment, prognosis and literature review. The importance of reporting this case refers to the rare incidence of paragangliomas in the nasal cavity and paranasal sinuses. Paragangliomas are neoplasias arising from paraganglionic tissue of the autonomic nervous system. Paraganglions are of neuroectodermal origin and present two types of cells: Type I, which contain catecholamine granules; Type II, which are supportive cells similar to Schwann cells surrounding type I cellsParaganglions are broadly distributed in the human body, found in the lungs, heart, mediastinum, gastrointestinal tract, retroperitoneal region and bladder. In the head and neck, they were found in trachea, tongue, larynx, hypophysis, pineal gland and orbit. Despite these findings, the most prevalent sites of paragangliomas are the carotid body, jugular body, along glossopharyngeal nerve and its tympanic branch, and the vagus nerve, especially next to nodal ganglionThis study reports a case of nasal paraganglioma in a 45-year-old male patient due to the rare aspect of such clinical condition at this specific site.Head and neck paragangliomas are very rare, with an incidence of 0.0012%The site of origin of nasal paragangliomas remains unknown. Many authors suggest the existence of paraganglionic tissue in the pterygopalatine fossa, based on paraganglions' close relationship with arteries and cranial nerves. However, among the cases previously reported in literature, the majority was found in the regions of ethmoidal middle concha or ethmoidal sinusJ.C., a 45-year-old Caucasian man searched for medical care complaining of nasal obstruction for one year. He also referred chronic nasal secretion with periods of purulent rhinorrhea. No bleeding, pain or facial edema was reported.th 2002, when total excision of lesion was carried out. Both the clinical pathology and final examinations confirmed the presence of a nasal paraganglioma. No immediate or late postoperative complications were observed. After 5 months of surgery, the patient was asymptomatic and attending outpatient follow-up. So far, he has not showed evidences of relapses.At otolaryngological examination, previous rhinoscopy showed a polyploid mass in the right nasal fossa. Fibroscopy revealed a lesion in the right nasal fossa obstructing the ipsilateral choanal portion. Computed tomography scan (CT) presented a lesion with dense soft tissues in the right nasal cavity . IncisioParagangliomas of the nose and of paranasal sinuses are very infrequent. According to a study comprising 73 cases of head and neck paragangliomas, only 3 were of nasal and paranasal typesNasal paraganglioma is a slow-growing neoplasia, with a time interval between symptoms' onset and diagnosis of 2 or more yearsClinical expression is recurrent episodes of mild to profuse epistaxis, rhinorrhea, nasal obstruction and facial edema, which may be followed by blurred visionzellballen which are separated by a rich capillary net of reticulin  combined with medullar carcinoma of thyroid gland and, optionally, as pleochromocytoma. In such cases, endocrine evaluation and magnetic nuclear resonance of the adrenal, thoracic and neck regions are necessary for an appropriate therapeutic strategyMalignant head and neck paragangliomas show an incidence of 4 to 19%The rare occurrence of paragangliomas of the nasal fossa has motivated our study. Once diagnosed, they should be treated surgically, considering its morbidity due to tendency towards progressive invasion of vital structures. However, even with adequate surgical approach, paragangliomas tend to present local relapse due to its nature and localization.


We encountered an extremely rare case of a vesicocutaneous fistula due to vesical diverticulitis with stones.A 78‐year‐old male patient presented to our department with complaints of suppurative discharge in the suprapubic area. Computed tomography revealed an enlarged prostate, a vesical diverticulum with stones located on the ventral side, and an aberrant connection between the anterior bladder wall and the external surface of the skin. The patient was diagnosed with a vesicocutaneous fistula due to vesical diverticulitis and was successfully treated with a multidisciplinary approach including vesical diverticulectomy with stone removal and nonviable tissue debridement. The patient continues to receive regular outpatient follow‐ups with urinary catheter changes.Vesicocutaneous fistulas due to vesical diverticulitis with stones are extremely rare. We should be aware that a vesical diverticulum with stones located on the ventral side might pose a high‐risk factor for the formation of a vesicocutaneous fistula in elderly patients. We report an extremely rare case of a vesicocutaneous fistula due to vesical diverticulitis with stones in an elderly patient. The case findings highlight the importance of considering the possibility of elderly patients developing vesicocutaneous fistulas due to a vesical diverticulum with stones located on the ventral side.Vesicocutaneous fistulas are abnormal tracts formed between the urinary bladder and the cutaneous surfaces of the body. Although rare, they have been reported after trauma, surgery, and radiation therapy on the pelvic area.A 78‐year‐old man presented to our department with complaints of suppurative discharge in the suprapubic area, intermittent dysuria, and a feeling of fullness in the lower abdomen for a few years.Staphylococcus aureus and viridans group streptococci. Computed tomography revealed multiple vesical stones (maximum diameter: 4.1 cm), and vesical diverticulum with stones located on the ventral side as well as an aberrant connection between the anterior bladder wall and the external surface of the skin, which was thought to be caused by the enlarged prostate  due to previously untreated chronic obstructive pulmonary disease with long‐term effects from smoking. On the other hand, the patient had no medical history that would predispose him to a neurogenic bladder. Blood tests revealed a mild inflammatory response . The pus culture test from the fistula revealed the presence of Vesicocutaneous fistulas are abnormal tracts formed between the bladder and the cutaneous surfaces of the body. These are caused by congenital diseases as well as complications of surgery and radiation therapy.We have summarized the clinical courses of 8 cases previously reported in the literature and that of our case in Table Surgical treatment is essential to cure this disease, and surgeries were performed in all reported cases. The purpose of the surgery is to control the infectious condition by removing the structures associated with the vesical stones and fistula. In the present case, the vesical diverticulum with stones were considered a contributing factor, and we performed multidisciplinary treatment including vesical stone removal, bladder diverticulectomy, and nonviable tissue debridement via an en bloc approach. Surgical wound dehiscence is one of the common complications after cystolithotomy. To prevent the occurrence of this complication, we cleaned the inside of the bladder with a large amount of saline, removed the infectious tissues, and retained as much normal tissue as possible.Vesical diverticulum formation occurs secondary to bladder outlet obstruction, which develops as herniations of the bladder wall between the defects of smooth muscle fibers.In conclusion, we report an extremely rare case of a vesicocutaneous fistula caused by vesical diverticulitis with stones in an elderly male patient. We should be aware that vesical diverticular stones located on the ventral side might pose a high‐risk factor for the formation of a vesicocutaneous fistula in elderly patients.Jun Akatsuka: Conceptualization; data curation; investigation; methodology; project administration; writing – original draft; writing – review and editing. Kyota Suzuki: Data curation; writing – original draft. Shunsuke Ikuma: Data curation. Masato Yanagi: Data curation. Yuki Endo: Data curation. Hayato Takeda: Data curation. Yuka Toyama: Data curation. Teruyuki Dohi: Data curation; investigation. Go Kimura: Supervision; writing – review and editing. Yukihiro Kondo: Supervision; writing – original draft; writing – review and editing.The authors declare no conflict of interest.The ethics committees of Nippon medical school hospital gave approval for this study (#30‐03‐1099) and written informed consent was obtained in accordance with the World Medical Association Helsinki Declaration.Written informed consent was obtained from the patient for publication of this case report and any accompanying images.Not applicable.


Emphysematous pyelonephritis, a necrotizing kidney infection, is a rare condition with a poor prognosis. We present a case of 24-year-old male with fever, malaise, and left flank pain. Computed tomography of abdominal and pelvis showed left emphysematous pyelonephritis. Treatment included IV antibiotics. A left nephrectomy was necessitated for definitive treatment. Clinical course after surgery was unremarkable. In conclusion, prompt diagnosis of emphysematous pyelonephritis is necessary to reduce morbidity and mortality. In some cases, nephrectomy is the definite treatment. Emphysematous pyelonephritis (EPN) is an acute severe necrotizing infection of the renal parenchyma and its surrounding tissue and leads to gas production in the renal parenchyma, collecting system, or perirenal tissue CT scan is mandatory not only to establish the diagnosis but also for follow- up and to decide the best line of treatment. Early diagnosis and management may facilitate renal preservation but delay in diagnosis and treatment contributes to high morbidity and mortality rates even today We describe a rare case of EPN in a 24-year-old male with poorly controlled, insulin-dependent type 2 DM.This case report was prepared in accordance with the SCARE guidelines A 24-year-old male with a history of poorly controlled, insulin-dependent type 2 DM presented to the ED with fever, malaise, left flank pain, and dysuria for 3 days. No other important past medical or surgical history. On exam, vital signs were: blood pressure 90/60 mm/Hg, pulse 140 bpm, and temperature 39.8°C. Physical examination showed delayed response to commands with mild disorientation. He had abdominal tenderness especially in the left flank. Examination of other systems showed no important findings.His laboratories are shown in E. coli which was sensitive to the meropenem and levofloxacin. Third day after surgery, we transferred the patient to the ward. He spent just one night and he was discharged on regular urological clinic visits. Follow-up for 6 months showed complete remission with normal blood tests including renal function and glucose level.Immediately, we started with fluid resuscitation with normal saline and we added insulin therapy to achieve target glycemic control. We sent urine specimen for culture. Abdominal and pelvis computed tomography (CT) scan revealed gas inside the left kidney . We starEPN is an acute severe necrotizing infection of the renal parenchyma and its surrounding tissue and leads to gas production in the renal parenchyma, collecting system, or perirenal tissue Diabetes mellitus is the single most common associated factor. Up to 95% of patients with EPN have underlying uncontrolled diabetes mellitus. Other reported factors associated with the development of EPN are drug abuse, neurogenic bladder, alcoholism and anatomic anomaly Plain abdominal radiography may show abnormal gas in the renal bed or collecting system. CT is the preferred diagnostic modality because of its high sensitivity and ability to assess the extent of gas patterns Treatment of EPN can be conservative with or without minor interventions or nephrectomy.The mortality rate of the disease ranges between 11% and 54% To our knowledge, this case is very rare because of the age of our patient (24 years old). Our case was a young male patient with poorly controlled DM type 2 diagnosed with EPN. The medical history and physical examination led to suspect EPN. We started first with fluid resuscitation. Empiric antibiotics were administrated via intravenous. CT scan of the abdomen and pelvis was the gold standard to confirm the diagnosis. Based on the clinical state of our patient, he underwent to simple left nephrectomy. The course after surgery was unremarkable. With strict control of glucose levels, he got full remission. Follow-up showed no complications.EPN is an emergency case. Even with young ages, it has a high mortality. Urologists should react immediately to deal with the case and give the suitable management for the patient.Written consent for publication of this case was obtained from the patient and is available upon request. Additionally, the patient received and approved this manuscript.


Burkholderia pseudomallei. The patient finally succumbed after multiple recurrences of the infection despite surgical excision of the osteomyelitic bone and the recommended antibiotic treatment. The management of cerebral venous sinus thrombosis in patients with cranial melioidosis is discussed along with a brief review of the literature.Cerebral venous sinus thrombosis is a rare complication of cranial melioidosis. We report a case of an adult male who presented with skull osteomyelitis, transverse sinus thrombosis and multiple brain abscesses. His blood cultures grew  Burkholderia pseudomallei [Melioidosis is an infection caused by the soil-dwelling Gram-negative organism, mallei . Infectimallei . CM can mallei . The casThis 51-year-old male patient with diabetes mellitus (DM) was admitted six times at our centre for treatment, the details of which are summarized in In the first admission, contrast computed tomogram (CT) of the brain showed imaging features suggestive of focal osteomyelitis of the left occipital bone near the transverse sinus  as seen in our case. The organism is known to cause scalp abscess , pachymeet al. was seen in our case [Involvement of multiple tissue layers from the scalp to the brain parenchyma as reported by Pit our case .et al. reported the first case of CM with CVT [The previously reported four cases of CM with CVT and the case we present are summarized in with CVT . Patientwith CVT . All patwith CVT . The comwith CVT . The othwith CVT . Our patwith CVT . Two prewith CVT  showed TDelay in diagnosis is a major concern in patients with melioidosis . The cliCultures from the CNS specimen is the best way to establish a definite diagnosis of CM . DiagnosThe appropriate surgical intervention followed by antibiotic therapy is the recommended treatment for CM. The antibiotic of choice and its optimal duration of treatment are controversial . VariousOf the four previously reported cases of CM with CVT, all had good outcomes . When CMClinicians should be aware of this rare presentation of simultaneous CNS and systemic infection in association with CVT in patients with melioidosis.Through this article, we recommend the following in patients with clinical presentation similar to ours (i) consider CM as one of the differential diagnoses in patients presenting with intracranial lesion, (ii) consider performing MR venogram in these patients as CVT can rarely be associated, (iii) consider long-term recommended antibiotic therapy and (iv) keep these patients on close clinical and radiological follow-up. If cranial imaging shows recurrence of infection and bacterial cultures remain negative, these patients should be treated again with antibiotics based on previous positive cultures for the recommended period of time.


ComprehGiven the low incidence of VTE in this population, extended chemical VTE prophylaxis is not routinely recommended, even in individuals who fall into a higher risk category; high BMI and multiple medical comorbidities . PostopeWe present the case of a patient with acute limb ischemia secondary to an arterial thrombus in the immediate postoperative period after an uncomplicated laparoscopic surgical staging for a stage IB grade 2 endometrioid EMCA.2The patient is a 63-year-old woman, gravida 4 para 2, with a past medical history significant for hypertension well controlled on Lisinopril, type 2 diabetes (HbA1 6.4%) controlled on Metformin, and class III obesity (BMI 42), who first presented with abnormal uterine bleeding, and was found to have FIGO grade 2 endometrioid EMCA on an endometrial biopsy. Pre-operative abdomen and pelvis computer tomography was performed that was only remarkable for a multi-fibroid uterus. She denied a history of bleeding or clotting disorders and did not have any significant cardiac or atherosclerotic disease. Her family history was unremarkable. Her past surgical history was pertinent for an exploratory laparotomy via a supra-umbilical horizontal incision after a trauma twenty years prior.Six weeks after her initial diagnosis, she underwent an uncomplicated total laparoscopic hysterectomy, bilateral salpingo-oophorectomy, and bilateral sentinel lymph node mapping and biopsy. She received 5,000 units of unfractionated heparin subcutaneously for peri-operative prophylaxis 30 min prior to surgical incision. Total surgical time including being in dorsal lithotomy position was 123 min. She underwent routine postoperative recovery and was discharged home on the same day.On postoperative day (POD) 1 she presented to the emergency room for sudden onset right foot pain associated with paresthesia and cold sensation. Bilateral lower extremity pulses were palpable, and the rest of the physical exam was found to be normal. Her labs were only significant for a magnesium level of 1.8 mg/dL, which was repleted orally. Her pain improved spontaneously without intervention, and she was discharged from the ED in stable condition.She presented to the emergency room again on POD 3 for worsening pain and paresthesia. Her physical exam was significant for new right calf tenderness on palpation, but otherwise remained unremarkable with palpable and equal peripheral pulses in both lower extremities. A lower extremity venous doppler was performed with no evidence of deep venous thrombosis. Labs were not collected during that visit. She was given one dose of 15 mg ketorolac intravenously and 10 mg cyclobenzaprine PO with some improvement in her pain. Her vital signs, electrolytes and creatinine were within normal limits, and she was discharge in stable condition.On POD 4, she presented once more to the emergency room for worsening pain in her right leg. On presentation, the right dorsalis pedis (DP) and posterior tibial (PT) pulses were not palpable, and absent flow was confirmed by bedside doppler. A computed tomography angiography (CTA) showed a thrombus extending 10 cm from the right external iliac artery down into the femoral artery, as well as an 8 cm thrombus in the right popliteal artery, confirming the diagnosis of an acute arterial thromboembolism . The pat313. In the medical literature, 9 case reports have previously described pelvic surgeries complicated by acute limb ischemia, 6 of which were in the setting of gynecologic malignancies  in lithotomy which was hypothesized to contribute to the occurrence of postoperative arterial thrombosis. The addition of head-down tilt during a case in lithotomy has also been proposed as a possible contributory factor by significantly decreasing lower limb perfusion intraoperatively . The patWhile the patient in question was similarly positioned intraoperatively, it remains unclear whether this was the sole cause of the postoperative arterial thrombosis formation. Furthermore, while our patient did not have a previous diagnosis of atherosclerosis, her CTA showed signs of early disease with mild calcifications noted in common iliac arteries. She also had a significant medical history of hypertension, diabetes and obesity as well as the diagnosis of EMCA, all of which are known to increase the risk of both arterial and venous thrombosis formation . Part of4As previously mentioned, acute limb ischemia secondary to arterial thrombi in the immediate postoperative period is extremely rare and not clearly understood. In analyzing the case at hand, we noticed a delay in timely diagnosis as the patient presented to the emergency department three separate times with worsening pain. High clinical suspicion for acute limb ischemia postoperatively is needed for prompt diagnosis and adequate treatment. Thorough clinical evaluation should include assessment of limb color, temperature, pulses, and motor and sensory function . ThromboFuture considerations for assessing patients at high risk for developing arterial thromboses can include evaluating whether ankle brachial pressure index needs to be routinely performed during pre-operative evaluation in patients with risk factors for atherosclerosis or in those with an existing diagnosis . AnotherThe authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.


Staged surgery strategy was preferred for patients with intra-extracranial communicating jugular foramen paraganglioma (IECJFP). A female patient who presented mild tinnitus, headache, and dizziness, together with preoperative related imaging, was diagnosed with a left intra-extracranial communicating jugular foramen lesion in November 2015 and accepted an initial operation for the intracranial tumor by retrosigmoid approach. The pathologic report was paraganglioma. In November 2021, a subtotal resection of the extracranial tumor was conducted for prominent lower cranial nerves (LCNs) deficit and middle ear involvement by infratemporal approach. In patients with IECJFP accompanied by LCNs deficit and middle ear involvement, an initial surgery for extracranial lesion and a second procedure for intracranial tumor were appropriate. However, the first operation for the intracranial lesion was preferred in IECJFP cases without LCNs deficit and middle ear involvement, as it could remove compression to the neurovascular structure and brain stem, clarify a pathological diagnosis, avoid a CSF leak, and prevent a severe neurological disorder from extracranial lesion excision. Subtotal resection of the extracranial tumor would be performed when lesion became larger combined with obvious LCNs disorder and tympanic cavity involvement. Consideration of specific staged surgical strategy for IECJFP in accordance with preoperative LCNs deficit and tympanic cavity involvement could prevent critical postoperative neurological deficit and improve quality of life in the long term. Paraganglioma is a kind of tumor rich in blood vessels, which can arise in different parts of the paraganglion system. Approximately 3% of extra-adrenal paragangliomas (PGs) were found in the head and neck, where PGs originated frequently from carotid bodies, jugulotympanic paraganglion cells and vagal nerves with low incidence at 1:30,000 in head and neck tumors . AlthougA 48-year-old female patient consulted a neurosurgeon due to mild tinnitus, headache, and dizziness in November 2015. A significantly enhanced intra-extracranial communicating jugular foramen lesion was found in the left enlarged jugular foramen on contrast-enhanced magnetic resonance images (CE-MRI), and attached to the brain stem and cerebellum  are commonly benign and steady tumors, but JFPs are also involved in important surrounding neurovascular structures . ErosionAccording to the Fisch classification for tympanic and tympanojugular paragangliomas , class DSurgery for IECJFP has been performed for several years, but incidence of serious postoperative complications was high . In spitJackson et al.  found thPostoperative LCNs paralysis of JFPs was more frequent than other PGs in the head and neck, whereas the rate of LCNs deficit was nearly 25%–50% . If preoWanna et al.  encouragRadiotherapy for JFPs was effective and feasible . ReferriWe could consider personalized staged surgical strategy together with noninvasive treatment to improve long-term quality of life in IECJFP patients with LCNs deficits and involvement of the tympanic cavity. Initial surgery for the extracranial tumor and a second procedure for the intracranial tumor are appropriate in IECJFP patients accompanied by LCNs deficit and middle ear involvement. However, for IECJFP cases without LCNs deficit and tympanic cavity involvement, the first operation for the intracranial lesion together with the second procedure with subtotal resection for the extracranial tumor would be preferred.


The typical feature is the autosomal dominant heritance and clinically dormant, non‐scarpalble, white diffuse, soft, thickened white plaques with a corrugated surface affecting mostly the buccal mucosa bilaterally which can substantially lead to the diagnosis of white sponge nevus. The typical feature is the autosomal dominant heritance and clinically dormant, non‐scarpalble, white diffuse, soft, thickened white plaques with a corrugated surface affecting mostly the buccal mucosa bilaterally which can substantially lead to the diagnosis of white sponge nevus. Intraoral examination revealed occlusal caries with respect to mandibular right first molar and was nontender, on percussion. A striking incidental finding was noted on performing her intraoral examination. There were asymptomatic, bilateral, white spongy plaques irregular in shape noted in the buccal mucosa, upper and lower labial mucosa, ventral surface of the tongue, and floor of the mouth Figure .There was no erythema associated with the plaques, but a generalized peculiar opalescent hue was present. On palpation, it was soft in consistency, nontender, and did not wipe off. The surface appeared smooth, thickened, folded, and corrugated with velvety texture. No lymph nodes were palpable. There was no relevant past medical history with no history of consanguinity. On further evaluation, the patient's mother reported similar lesions in her oral cavity. On examination, similar lesions were noted with respect to bilateral buccal mucosa and ventral surface of the tongue in the 36‐year‐old mother Figure .The patient and her mother both were asymptomatic but reported a history of irregular use of topical antifungal agents (prescribed by dentists) since the initial presence of the lesion with no improvement or reduction in the size of the lesion to date. They were not worried about the lesion as according to them they have been living with it since childhood, also the mother reported similar lesions in her mother. Patients were not willing for a biopsy, so it could not be carried out. Moreover keeping in mind the insignificance contribution of biopsy to the treatment plan, the cost, and trauma involved, the diagnosis of WSN was given, which was purely on the basis of history and clinical findings of the duo. The patients were referred to the Department of Ophthalmology and Obstetrics/Gynecology to rule out the presence of conjunctival and genital lesions. Therapeutic management was not needed as the lesions were asymptomatic.3White sponge nevus is considered a rare genetic disorder, affecting 1 in 200,000 people.The characteristic clinical presentation of WSN is that of white plaques in the oral cavity, most predominantly affecting the buccal mucosa.Prominent hyperkeratosis, marked acanthosis, spongiosis, cleared cytoplasm with perinuclear eosinophilic condensation are a common histological characteristic feature.The differential diagnosis includes a diversity of processes  Table .Based on the history, clinical presentation, and after ruling out other white lesions, we arrived at the diagnosis of WSN.To date, only 1 case of malignant transformation has been reported which was attributed to the chronic use of prednisone.4In conclusion, first the attending clinician should try to establish etiopathologic correlations with the list of processes in differential diagnosis and then consider performing a biopsy to confirm the diagnosis and also to rule out possible malignancy, particularly for patients with history of use of tobacco product and/or alcohol. If a patient is asymptomatic and does not desire any treatment or is unconcerned about the lesion, we strongly recommend to keep the patient on regular follow‐up for any changes. This case report is presented for its rarity and also its resemblance to other white lesions. Hence, it is the duty of an oral physician to correctly diagnose the condition to avoid unnecessary treatment.Abhishek Gupta: Conceptualization; formal analysis; methodology; project administration; resources; supervision; validation; visualization; writing – original draft; writing – review and editing. Ram Sudhan Lamichhane: Conceptualization; formal analysis; supervision; validation; writing – review and editing. ANJU REDHU: Conceptualization; formal analysis; supervision; writing – original draft; writing – review and editing.There were no sources of funding.The authors declare that there is no conflict of interests regarding the publication of this paper.Written Consent was taken from the patient and patient's guardian.


The study aimed to observe the efficacy of the different common approaches used by a single neurosurgeon. All the patients were evaluated preoperatively and during follow-up with campimetry, head CT scan, and post-contrast MRI. (3) Results: After a transcranial approach, visual acuity improved in 86.20%, was stable in 10.34%, and deteriorated in 3.45%. Through transsphenoidal surgery, vision improved in 80%, was static in 20%, and deteriorated in 0%. Transcranial approaches included pterional, mini-bifrontal basal, and supraciliary keyhole microscopic craniotomies. Gross total removal was performed in 58.82%, near total in 10.34%, and partial removal in 3.45%. The transcranial/supraciliary keyhole endoscopic-assisted approach showed a gross total removal rate of 80%, and near total in 20%. The transsphenoidal approach showed a gross total removal rate of 60%, near total in 20%, and partial removal in 20%. (4) Conclusion: Endoscopic-assisted keyhole supraciliary mini craniotomy for resection of tuberculum sellae meningioma offers low morbidity and good visual outcome. The endonasal route is preferred for the removal of TSM when they are small and midline placed. The major limitation of this approach is a narrow surgical corridor and the restriction on midline-placed lesions. Gross total removal was better achieved with mini-bifrontal basal and pterional craniotomies. Tuberculum sellae meningiomas (TSM) account for 5–10% of all intracranial meningiomas and typically arise from the dura mater of tuberculum sellae, chiasmatic sulcus, and limbus sphenoidale ,3,4,5. VThis is a retrospective study aimed to observe the efficacy of the different common approaches used by a single neurosurgeon. The approaches were a minipterional approach, a supraciliary keyhole microsurgical approach, an endoscope-assisted supraciliary keyhole approach, a bifrontal basal approach, and an extended endoscopic endonasal approach in resecting the tuberculum sellae meningioma. All the patients were evaluated preoperatively by campimetry and postcontrast MRI. Clinical postoperative follow-up included visual perimetry tests at one month and radiologically by postcontrast MRI at one and at six months. In some cases, due to the lack of economic resources, postcontrast CT head scans were performed.The patient was positioned supine, with the head turned about 30° to the opposite side along the sphenoid wing and slightly (15°) retroflexed. The head was fixed in a three-pin Mayfield head holder. The scalp incision started just above the tragus, anterior to the superficial temporal artery, continuing superiorly behind the hairline and ending near to midline. Subgaleal and interfascial dissection was conducted to preserve the frontal branch of the facial nerve. The temporalis muscle was incised down to the periosteum, beneath the skin incision, and then the muscle and the pericranium were reflected anteriorly and inferiorly. The McCarty keyhole was used as a starting point for the craniotomy. The bone flap was cut and elevated. The dura mater was opened in a curved fashion with the sphenoid wing on its base. The next step was splitting the sylvian fissure to drain the cerebrospinal fluid. A representative case of our case series is presented below.A 55-year-old woman presented with gradual left eye blindness and occasional headaches. A brain MRI with Gadolinium revealed a TSM associated with left optic canal invasion. She underwent left sided minipterional craniotomy. Optic canal unroofing and anterior clinoidectomy were performed, and the tumor was devascularized from its attachment from the tuberculum sellae. The optic–carotid triangle area was exposed. The tumor was removed in piecemeal fashion, through the pre-chiasmatic and optic–carotid space. Gross total removal of the tumor was achieved. Her post-operative period was uneventful, and her vision improved to finger count .The patient was placed in the supine position with the head turned 15–20° to the contralateral side and 15° retroflexed to allow the frontal lobe to slightly fall back. The head was fixed using the three-pin head holder. A curvilinear skin incision was made, and then one burr hole was placed at the McCarty’s point. The craniotomy was made in a standard fashion, of about 2 cm in length and 2 cm in height. If the frontal sinus was exposed intentionally or accidentally, the sinus mucosa was removed and cauterized, and the sinus was sealed with autologous and/or heterologous tissue. The dura was opened in a semilunar fashion with its base towards the orbital rim. The frontal lobe was slightly retracted to identify the suprachiasmatic cistern and opened to drain the CSF to give more brain relaxation. Arachnoid opening of the sylvian fissure started from medial to lateral. The opening of the sylvian fissure further facilitated the dissection and the anatomic exposure, revealing the major structures of the peritumoral area. The next step was to devascularize the tumor at its basal attachment. Tumor mass debulking was conducted with microsurgical technique.A 45-year-old lady presented with a bitemporal field deficit along with mild headaches. Her brain MRI revealed the presence of a tuberculum sellae meningioma. She underwent keyhole supraciliary mini craniotomy and microsurgical removal of tuberculum sellae meningioma. Her postoperative recovery was excellent with visual outcome improvement .This approach is like the supraciliary keyhole microsurgical approach, but here we used a 0° endoscope . The endoscope was held by an endoscopic holder , and the tumor dissection was carried out using micro instruments and micro scissors. Long bipolar forceps were used for cautery. The patient was placed in the supine position with the head turned 15–20° to the contralateral side and 15° retroflexed to allow the frontal lobe slightly to fall back. Patient position, supraorbital incision, and the size of craniotomy were like in the supraciliary keyhole microsurgical approach. Here, CSF was gently sucked out from the basal cistern to make frontal lobe slack, and the endoscopic micro instruments were manipulated in between the space of frontal bone and frontal lobe.A 35-year-old woman presented with a bitemporal field defect. Head MRI revealed TSM. She underwent an endoscopic-assisted supraciliary keyhole approach. The tumor was resected in piecemeal fashion. Her recovery was excellent and visual improvement was satisfactory .With the patient in a supine position, the head was fixed in a three-pin head holder. A midline osteotomy was performed, extending close to the orbital roof. Two burr holes were placed on the orbital bottoms on each side through a bicoronal skin incision beyond the hair line. The dura was opened parallel to the base and the sagittal sinus was ligated and cut at the cecal foramen, followed by transection of the falx, preserving the bridging veins. The frontal poles were retracted gently under magnification, using two self-retaining retractors adjusted stepwise. The interhemispheric and the bilateral olfactory cisterns were opened to drain the CSF. We investigated two cases using the bifrontal basal approach.A 56-year-old woman presented at our institution complaining about an impaired level of consciousness and a prolonged history of headaches and visual disturbances. Brain MRI revealed a huge tuberculum sellae meningioma with diffuse perilesional edema. She underwent an emergency minibifrontal craniotomy and tumor removal in piecemeal fashion. Her recovery was good, and her vision improved .A 0° endoscope, 4 mm in diameter , was used freehand. Once the endoscope had been inserted into the chosen nostril, the inferior and middle turbinates, and the nasal septum were identified. Using Tilley forceps, long cotton pledgets, soaked in diluted adrenaline  or xylometazoline hydrochloride, were inserted in the space between the middle turbinate and the nasal septum to achieve a vasoconstrictive effect of these richly vascularized structures. The middle turbinate was gently medialized or removed to make sure that the surgical corridor that passes between the nasal septum and the turbinate itself was wide enough. Once the cotton pledgets were removed, adequate inspection of the posterior portion of the nasal cavity was conducted, and the choana, the sphenoethmoidal recess, and the sphenoid ostium were identified. After a careful inspection, a nasoseptal/Hadad flap was taken starting from the choana to the floor of the nasal septum, then turned upward near to the roof of nasal septum. The Hadad flap was parked into the nasopharynx.The sellar floor was drilled and opened initially using a small diamond drill, and then enlarged with Kerrison bone punches. Once inside the sphenoid, the key anatomical landmarks were identified, including the planum sphenoidale, tuberculum sellae, sellae, lateral optico-carotid recesses (OCRs), optic canals, and clinoidal carotid protuberances. The bone from the mid-to-upper sellae was removed with a combination of the diamond drill and the Kerrison punches. Next, the bones of the tuberculum sellae and planum were removed. This is often hyperostotic and could be vascularized due to the adjacent tumor feeder from the ethmoidal arteries. The use of a diamond burr is safer than a cutting burr when working around critical neurovascular structures and is also helpful for bony hemostasis. The bone overlying the proximal optic canals would be unroofed to access the tumor extension into the medial optic canals bilaterally. Drilling with continuous irrigation should be performed to avoid thermal injury to the optic nerves. The lateral OCRs help estimate the location of the canalicular segment of the optic nerve. A key to achieving wide access to the optico-carotid cistern was to remove the lateral strut of the tuberculum sellae. The dura was then incised as cruciate or in a rectangular fashion. The tumor was removed in piecemeal fashion and bleeding was secured. Surgicel was used as a hemostatic agent 44. The gasket seal technique was used to close the dural defect, aided with fibrin glue. Merocel nasal pack was introduced as a mucosal hemostat.A 45-year-old lady was presented with bilateral visual impairment and occasional headaches. A head MRI revealed a small-size TSM. She underwent an extended endonasal approach and gross total removal of the tumor. Her vision improved significantly in the post-operative period .Data were collected from 2015 to 2020 and performed by a single operator. We retrieved 34 patients affected by TSM. Patient’s age distribution ranged from 11 to 65 years. Most of the patients were aged between 31 to 50 years (61.76%) . Among tTSMs are relatively common and are a formidable surgical challenge ,20,21,22We prefer this approach when the TSM has significant extension towards the optic canal and the optic–carotid recess to the parasellar area. In this case, we need to accomplish an optic foramen decompression to remove the meningioma from the optic canal and remove the tumor lateral to the optic nerve and carotid artery. A minipterional craniotomy and a transylvian approach provide a favorable corridor to deal with optic foramen and the optic–carotid recess area.This technique is a medial approach to find the tuberculum sellae meningioma. We choose this approach for small meningiomas, midline located, and with no or minimum ICA involvement or displacement.We prefer this technique when the TSM is extended to the sellae and mainly occupies the sellae, while directing posteriorly with no or less A-Com complex encasement. It gives us direct exposure to the sellae and tuberculum sellae. We use a three-hands approach where a 0-degree endoscope is held by an endoscopic holder or by an assisting surgeon. The operator works bimanually with the endoscope and dedicated tools.We choose this approach when the TSM is greater than 4 cm, with perilesional edema and partial or total obliteration of the frontal horn of the lateral ventricle. The neck is needed to be hyperextended to achieve better exposure of the tumor.We prefer this approach when the TSM is extended to the sellae, occupying the sellar region, directing posteriorly towards the third ventricle with no or minor A-Com complex encasement. We perform a three-hand approach where a 0-degree endoscope is held by an assistant surgeon and the operator works bimanually.Younger patients tolerated the surgical procedure better than older patients, in which a more conservative resection associated to complementary radiosurgery should be considered in the surgical planning. Systemic factors, such as hypertension and diabetes mellitus, could affect the surgical procedure and outcome to a certain degree. The extent of visual deficit was the single most important factor that determined the course of surgery. The entity of the involvement of the optic nerve and of the associated visual deficit is strongly related with a tighter relationship of the neurovascular structures  with the tumor, and consequently with a more difficult dissection. Visual symptoms arise early and usually are slowly progressive ,31,32,33Endoscopic-assisted keyhole supraciliary minicraniotomy for resection of tuberculum sellae meningioma offers low morbidity and good visual outcome. The endonasal route is preferred for the removal of TSM when they are small and midline placed. The major limitation of this approach is a narrow surgical corridor and the restriction to midline-placed lesions. Gross total removal was better achieved with minibifrontal basal and pterional craniotomies. The arachnoid surrounding the optic nerves, chiasm, and anterior circulation artery must be spared to improve the visual function postoperatively.


Ectopic thymic tissue in the subglottis is an extremely rare disease that causes airway obstruction. Few cases reported were accurately diagnosed before surgery.A case of a 2-year-old boy with airway obstruction caused by a left subglottic mass was reported. The presentation of radiological imaging, direct laryngoscopy and bronchoscopy, pathology, and surgical management were reviewed. An extensive search in PubMed, EMBASE, Web of Science, Google Scholar, and EBSCO of English literature was performed without a limit of time.Besides our case, only six cases were reported since 1987. The definitive diagnosis on these patients were made with the findings of pathology, of which, five were ectopic thymus and two were ectopic thymic cysts. Our case was the only one with a correct suspicion preoperatively. Four cases underwent open surgical resection, and two cases underwent microlaryngeal surgery, while one deceased after emergency tracheostomy. No recurrences were found by six patients during the follow-up after successful treatments.Ectopic thymus is a rare condition, infrequently considered in the differential diagnosis of subglottic masses. Modified laryngofissure may be an effective approach to removing the subglottic ectopic thymus and reconstructing the intact subglottic mucosa. Ectopic thymic tissue in the subglottis is an extremely rare disease, which causes airway obstruction. According to the definition, aberrant cervical thymus denotes the presence of thymic tissue in any area  outside its normal descent path.We undertook a review of literature published in English language with focus on ectopic thymus of the subglottic area. Databases searched in this study included PubMed, Embase, Web of Science, Google Scholar, and EBSCO. The literatures search was not limited by time. Search terms included ectopic thymus, subglottic stenosis, subglottic mass, and laryngeal and subglottic mass. Reference lists of the found articles were scanned for other case reports.A 2-year-old boy with recurrent stridor and multiple episodes of acute laryngitis and progressive symptoms of upper airway obstruction was admitted to our department in June 2020. General physical examination showed normal lung and heart function and no signs of respiratory tract infection. No palpable neck mass was found. He had no swallowing difficulty or hoarse cry. Normal vocal cord motion was identified by flexible fiberoptic laryngoscopy.Abnormal signal was found at the thyroid level on neck Magnetic Resonance Imaging (MRI) scan B–D; the Direct Laryngoscopy and Bronchoscopy (DLB) examination under general anesthesia was performed, revealing a subglottic semicircular mass with smooth surface mucosa and obstruction of 70% of the subglottic cavity . ModifieThe diagnosis of ectopic thymus was confirmed by pathohistological evaluation of the biopsy of the subglottis. Other possible etiologies  were excluded based on the patient’s medical history, clinical presentation, laboratory and microbiological examination, and imaging studies. Postoperative pathohistological examination showed Hassall corpuscles . The diaDLB was performed after removing ETT one week after the surgery. DLB showed bilateral aryepiglottic fold congestion and edema, but a patent subglottis. Mucosal healing was good, and no abnormal secretion was observed. The patient was transferred to the general ward. According to the results of sputum bacterial culture and drug sensitivity, anti-inflammatory treatment was given, followed by budesonide nebulization and anti-reflux treatment. Laryngeal obstruction was significantly relieved. No recurrence was found during two-month follow-up.Cervical ectopic thymus is a rare form of congenital neck mass in children.Theoretically, an ectopic thymus could occur in any other location outside the normal descend pathways, such as pharynx, trachea, posterior neck or mediastinum, or esophagus,Normally, thymus tissue originates from the ventral development of bilateral third pharyngeal sacs during the sixth week of embryonic development.Nowadays, mainstream view of the occurrence of subglottic thymus is that could be a result of the failure descent of thymic in the embryo. If thymic precursors are pulled medially before cricoid cartilage formation at seventh week, it may be trapped within the annulus.If a mass in the neck or anterior mediastinum is suspected of an abnormal thymus tissue, ultrasound is the first choice for the evaluation. In the published cases,21CT scan as the criterion standard for radiological diagnosis could be suggested when ultrasound results cannot provide a hint to make a definitive diagnosis.Some authors believe that MRI scanning is the best non-invasive method to identify abnormal solid thymic problems. MRI of the normal thymus shows uniform signal intensity or slightly higher intensity compared with the muscles on T1-weighted images, and the signal intensity close to that of fat on T2-weighted images.The present preoperative examination could not accurately identify the subglottic mass as ectopic thymus tissue. Owing to these limitations and rarity, six cases of ectopic subglottic thymus previously reported in the literatures were misdiagnosed preoperatively. Indeed, a correct diagnosis can be made only by pathohistological examination of the excised specimen. Hassall corpuscles, which are restricted to the medulla and characterized by a concentric pattern of keratinization, are the most typical feature of thymus.The broad differential diagnosis of pediatric neck masses typically fits into three main categories: inflammation, tumors, or structural abnormalities. There is cervical lymphadenitis, branchial arch cysts, lymphatic malformations, infantile hemangioma, squamous papilloma, parathyroid adenomas, dermoid or epidermoid cysts, carcinoids, and mucoepidermoid carcinomas in common causes.For differential diagnosis, imaging examination and pathohistological evaluation of the biopsy are required. Clinic radiological approach based on morphologic features is useful for evaluation of a suspected thymic mass. DLB examination should be performed to visualize the mass for proper differential diagnosis.The heterotopic subglottic thymus may cause recurrent respiratory distress and stridor. Although these symptoms can be partially alleviated by drug therapy, surgery is the gold standard for the purpose of both diagnosis and treatment, especially in cases of any symptomatic subglottic mass. A complete excision of the ectopic thymus is also important, as cases of malignant degeneration have been reported.In previous reports, several surgical approaches were used for the excision of ectopic thymus. In Case 1, under the condition of tracheotomy, a relatively unvascularized mass was found between the left superior pole of the thyroid gland and the trachea, extending backwards to the tracheoesophageal sulcus, which was completely removed. In Cases 2 and 4, thymic cysts were removed using a microlaryngoscopic surgery with a combination of cold instruments and microdebrider. No clear boundary was found between normal mucosa and the subglottic mass, and therefore marsupialization was deemed sufficient. But Case 4 repeated endoscopic surgery for the recurrence. In Cases 3 and 6, transverse neck incisions were performed to fully expose the trachea, cricoid cartilage and thyroid cartilage. Vertical midline incision was made through the cricoid cartilage to fully expose the subglottic lesions. Temporary tracheostomy or ETT inserted in the lower airways was performed for adequate exposure of the subglottic lesion in Case 3. In our case, we conducted a standard transverse neck incision, opening the cervical fascia vertically along the midline, preserving the strap muscles, and allowing a good exposure of the anterior thyroid and cricoid cartilage and the first two or three tracheal rings. The cricoid cartilage was cut vertically along the midline up to 1 cm below the vocal fold plane. An obvious mass between the perichondrium of the cricoid cartilage and the mucosa of the subglottic trachea was fully resected without tracheostomy. The intact subglottic mucosa was reconstructed to avoid the risk of postoperative stenosis.The presence of mediastinal thymus should be confirmed before surgical resection of subglottic mass, because theoretically the removal of the thymus tissue may cause mental abnormalities, affect the development of normal immune function or induce autoimmune diseases.27In the six published cases, no recurrences were found during the follow-up. In our case, a planned DLB was performed two months later, and no recurrent airway lesions were found.For children presenting with subglottic mass, the possibility of ectopic thymus should be considered in the differential diagnosis. To best of our knowledge, this is the seventh case in the English literature and the only one with a correct suspicion prior to surgery. Modified laryngofissure may be an effective approach to removing the subglottic ectopic thymic and reconstructing the intact subglottic mucosa. Pathohistological evaluation of the biopsy is the gold standard for diagnosis. The knowledge and awareness about the subglottic thymus entity enable the surgeon to make accurate diagnosis and treatment.The authors declare no conflicts of interest.


Nivolumab, an anti-PD-1 check point inhibitor, is an immunotherapeutic agent, representing a major step in the treatment of melanoma. However, its use is associated with severe toxicities. Among them, gastrointestinal (GI) disorders from the lower GI tract have been widely reported. On the contrary, disorders from the upper GI tract are rare. Such a case of delayed nivolumab induced severe gastritis in a 53-year-old Caucasian female patient suffering metastatic melanoma is described. The patient’s symptoms from the upper GI tract began 4 months after nivolumab treatment initiation. The diagnosis was based on imaging, including PET/CT, endoscopical and pathological findings. The side effect was successfully treated with prolonged administration of proton pump inhibitors and corticosteroids. There are only a few cases of immune check point inhibitors (ICPis) induced upper GI tract disorders, while it seems that the symptoms from nivolumab induced upper GI tract damages appear later than those reported in the lower part. Nivolumab, among other side effects, may cause severe gastritis. Hence, this pathological entity should be included in the list of this drug’s side effects. Immunotherapy represents a major step in the management of melanoma, since it has improved dramatically responses to treatment and overall survival of patients suffering this malignancy. However, this treatment may be associated with severe, sometimes life threatening, toxicities ,2.Immune suppression caused by malignant cells and consequent evasion of healthy tissues are hallmarks of the cancer metastatic process. A series of co-inhibitory and co-stimulatory receptors, and their ligands, are involved, controlling these mechanisms such as the programmed cell death protein-1 (PD-1) and/or the programmed death-ligand-1 (PDL-1) axis, that stand out as therapeutic targets .Nivolumab is a human IgG4 monoclonal antibody against PD-1, acting as an immune check point inhibitor (ICPi). ICPis block a signal, given by the tumor cells, that prevents activated T-cells from killing the malignant ones, thus allowing the immune system to attack the tumor. Nivolumab has been widely used successfully as monotherapy or in combination with other ICPis against several malignancies including melanoma [The use of nivolumab, however, has been associated with a number of side effects, such as fever, immune related inflammation and dysfunction of several organs, namely pneumonitis, colitis, hepatitis, nephritis, hypo-and hyper-thyroidism, type I diabetes mellitus, iridocyclitis, neuropathies and skin toxicities. This drug’s gastrointestinal (GI) toxicities have been often observed and widely reported. However, gastritis has only recently been included in the reported GI toxicities of this agent ,6. HenceThe case of an extremely rare nivolumab induced severe gastritis in a 53-year-old Caucasian woman with metastatic melanoma (MM) is presented. The role of PET CT in diagnosing this rare complication is also reported.A 53-year-old female patient suffering MM was hospitalized for progressive and gradually deteriorating loss of appetite, gastric discomfort and pain, nausea and vomiting during the two previous months, with consequent weight loss.She had prior history of pharmaceutical iridocyclitis of the right eye and hypothyroidism, due to side effects of the BRAF and MECK inhibitors combination treatment she had received for her MM disease. At that point in time, she was on nivolumab monotherapy every two weeks, already for 6 months.Her clinical examination, on admission, was unremarkable, except for mild sensitivity at the deep palpation of the gastric area. Her hematological and biochemical profiles were within normal limits.A PET/CT, performed soon after her admission to the hospital, to evaluate the MM status and to restage her disease, showed intense metabolic uptake in the stomach area, indicating a malignant entity such as infiltration by MM, lymphoma or gastric carcinoma. Inflammation was also included in the differential diagnosis, taking into account the intense glucose metabolism of inflammatory cells  Figure .A consequent esophago-gastro-duodenoscopy (OGD) revealed severe erythematous gastritis, with frangibility, edema and mucous exudate, involving the antrum and the duodenal bulb with no ulcers seen macroscopically. Biopsies have been taken from the gastric mucosa.Histology of the biopsy specimens of the gastric and duodenal mucosa have shown moderate inflammatory infiltration of the lamina propria, increased number of eosinophils in the lamina propria and extension of the eosinophils into the surface epithelium, consistent with the diagnosis of severe gastritis .She was started on proton pomp inhibitors (PPIs) and prednisolone at a dose of 0.5 mg/kg for 10 days with gradual improvement of her symptoms, starting six days after the initiation of this treatment.She continued to receive PPIs and prednisolone in progressively decreasing doses for 3 months, with further, and finally complete, resolution of the symptoms from the upper GI tract.A new gastroscopy performed three months later, while the nivolumab treatment had been discontinued, did not reveal signs of gastritis.A rare case of nivolumab-induced gastritis with its treatment and outcome is presented. The diagnosis has been based on endoscopical and pathological findings, while imaging (PET-CT), although is not included in the standard diagnostic process of gastritis, also indicated this rare complication in the present case.Cumulative data from anti PDL-1/PD-1 treatments’ experience have shown that side effects from the GI tract occur often, reaching an incidence of 26% [However, there are only a few case reports of ICPis induced upper GI tract disorders. In particular, there is a very limited number of nivolumab-associated upper GI tract toxicities reported in the English literature. Boike et al. reported esophagitis and gastritis after nivolumab treatment for malignant lymphoma, Kobayashi et al. hemorrhagic gastritis after nivolumab treatment for non-small cell lung cancer, Nguyin Tran et al. nivolumab-associated upper GI injury, Ojega Gomez et al. nivolumab induced neutrophilic gastritis, while Vindum et al. severe steroid refractory nivolumab induced gastritis, finally treated with infliximab ,9,12,13.The present patient’s gastroscopy revealed erythematous gastritis, severe edema and mucous exudate with no ulcers, while histology of the gastric mucosa has shown remarkable infiltration by eosinophiles. In particular the mucosa had normal architecture and mucin production. There was an inflammatory infiltration of the lamina propria with an increased number of eosinophiles, while eosinophiles were also extended into the surface of the epithelium. It is of note that other investigators have observed similar histological inflammatory findings induced by nivolumab and other ICPis with infiltration of the gastric mucosa by eosinophiles and also by neutrophils and/or by CD4, CD8 and CD20 lymphocytes ,14.PET/CT contributed, in this case, to the diagnosis of this extremely rare drug-related complication. In general, PET/CT has demonstrated to possess high diagnostic performance for the detection of soft-tissue, nodal and visceral metastases at initial staging or during follow-up, while this imaging examination represents a valuable tool for identification of tumor responses early in the course of several antineoplastic treatments. Furthermore, regarding immunotherapy, PET/CT has demonstrated the capability of assessing tumor response on a whole-body basis and detecting signs of immune activation as well as immune-related adverse effects ,16. In tIt should be noted that even though PET/CT was used in the reported case, the diagnosis of gastritis is made through medical history and physical examination, while these findings are confirmed with esophago-gastro-duodenoscopy. The PET/CT was performed soon after the patient’s admission to the hospital in order to evaluate the MM status and to restage her disease.ICPis associated lower GI tract toxicity, such as colitis, appearing six to nine weeks after initiation of treatment, has been widely observed and reported ,2,6. On This observation, along with the few previous reports, permits the assumption that symptoms from the upper GI tract nivolumab induced damages appear later than those reported of the lower part ,2.The mechanisms underlying these immune response adverse events remain unknown. It has been proposed that cell and tissue injury is due to self-reactive CD8 -positive T cells, and/or that the damage is probably caused by autoantibody production from CD4-positive T cells .Steroids are the treatment of choice for the ICis induced GI toxicities, whereas the anti-tumor necrosis factors antibodies, infliximab and vedolizumab have been successfully used in severe and steroid-refractory gastritis and colitis cases ,9,13.Probably the incidence of autoimmune induced gastritis might be underestimated, since gastroscopy is not routinely performed in cases with upper GI symptoms such as nausea, vomiting, odynophagia, dysphagia and epigastric pain.The present patient responded promptly and well six days after initiation of PPIs and steroid treatment, which was continued, in lower doses, for a prolonged time period.In conclusion, nivolumab, among other side effects, may cause severe gastritis. Hence, gastritis should be included in the list of this drug’s side effects. Oncologists should have an increased index of suspicion for this complication, in patients treated with nivolumab. Even though PET/CT contributed to the diagnosis of the reported case, it should be noted that esophago-gastro-duodenoscopy represents the gold standard diagnostic approach for confirmation of this particular and severe side effect.


Bilateral choanal atresia is a surgical emergency because of the risk of neonate death from acute asphyxia if treatment is delayed. Its diagnostic confirmation is often endoscopic or CT scan and requires a search for associated malformations.We present the case of a nine-year-old girl who was referred to the ENT department with suspected adenoid pathology. Her medical history showed respiratory distress at birth treated as a neonatal infection. We suspected bilateral choanal atresia due to the absence of fogging on mirror test and failure to pass a 6Fr or 8Fr suction catheter through the nasal cavity into the nasopharynx. Facial CT confirmed the presence of bilateral mixed osteo-membranous choanal atresia. Transpalatal choanoplasty was successfully performed with pre and postoperative endoscopic examination.This clinical case adds to the limited literature on bilateral choanal atresia diagnosed long after birth, raising once again the lack of knowledge of choanal atresia by some health workers, emergency neonatal care, the mechanism of breathing in the newborn, and the management of this malformation. Transpalatal choanoplasty is a good alternative when technical conditions do not allow an endoscopic endonasal approach. Choanal atresia (CA) is a rare birth defect characterized by imperforation of the posterior part of one or both nasal cavities. Neonates are obligate nasal breathers making bilateral CA a surgical emergency requiring prompt management for airway control. Its diagnosis is clinical, sometimes requiring endoscopic or CT confirmation and a search for associated defects. The treatment of CA is surgical and can be performed either as an endonasal or endobuccal procedure. Several surgical approaches have been described  but transnasal and tranpalatal are commonly used . RecurreA nine-year-old girl was referred to our department for a suspected adenoid pathology. Clinical history reported nasal obstruction and chronic bilateral mucosal rhinorrhea since the first weeks after birth. Birthing history revealed that the mother had been followed in a peripheral care unit and no gravidic or non-pregidic pathology was noted. At birth, respiratory distress was reported and was treated as a neonatal infection for a two-week period. No measures to secure the airways had been reported. From then on, the evolution was marked by mouth breathing, breathing difficulties during feeding, restless sleep with apnea episodes, and delayed growth in height and weight. The ENT physical examination noted a weight of 21 kg, mouth breathing, no fogging on the mirror test, and a positive rubber catheter test. Bilateral choanal atresia was strongly suspected. Facial CT confirmed bilateral mixed osteomembranous choanal atresia , cerebral malposition , congenital endonasal masses , embryonic tumors, or arhinia ,6. With Due to the high position of the larynx in newborns, breathing through the mouth is almost impossible. Breathing through the mouth is obstructed due to the fact that the epiglottis is very close to the soft palate and that tongue is in close contact with both the soft and hard palate in the neonatal period . In bilaThe care of bilateral CA requires emergency medical procedures such as placement of an adapted Guedel’s cannula or orotracheal intubation, diagnostic confirmation by nasal endoscopy and/or craniofacial CT scan, and then prompt surgical care. In our resource-limited settings, direct endoscopic exploration under general anesthesia upon strong clinical suspicion could help avoid CT scans and reduce the cost of treatment, which is often borne by patients.Surgically, the endonasal and transpalatal approaches are the most frequently used, but the nasal endoscopic approach is increasingly preferred in choanoplasty. Because of the lack of all the endoscopic surgical equipment adapted to our patient who was nine years old and 21 kg, the transpalatal approach was used successfully with pre and postoperative endoscopic controls. Restenosis, the most frequent complication of CA cure, is possible whatever the surgical technique used. Nasal cavity stenting does not seem to avoid the risk of restenosis according to some researchers and its impact on the surgical outcome is still a matter of controversy ,9. AdjuvBilateral CA is a rare and serious defect due to the risk of neonatal asphyxia. Adherence to a newborn care protocol, including systematic verification of nasal cavity patency, could help prevent at worst some neonatal deaths and late diagnosis of CA at best. Transpalatal choanoplasty appears to be a good alternative when technical conditions do not allow an endonasal endoscopic approach.


We presented the 16th documented case of cavernoma with TN. Although cavernoma involving the trigeminal nerve is extremely rare, this diagnosis should be taken into consideration when a lesion in the cerebellopontine angle is detected on MRI, and the clinical manifestation is consistent with that of secondary TN. Specialized MRI sequences, such as susceptibility weighted imaging (SWI), gradient echo T2, and constructive interference in steady-state (CISS)-weighted imaging, aid in establishing the diagnosis. Resection via craniotomy may be the primary management strategy for cavernoma causing TN. In addition, gamma knife radiosurgery (GKRS) and percutaneous balloon compression (PBC) may ameliorate the pain to some extent.Cavernoma is the second most common cerebrovascular lesion. Cavernoma involving the cranial nerves is very rare. Only 15 cases of cavernoma presenting with trigeminal neuralgia (TN) have been previously reported. Here, we report a rare case of cavernoma manifesting with TN. A young female patient with a 15-day history of right-sided lancinating pain in the face, difficulty in opening the mouth, and hearing dysesthesia. Magnetic resonance imaging (MRI) revealed a well-demarcated lesion in the cerebellopontine angle related closely to the root of the trigeminal nerve. The initial impression was that of a neurinoma. The lesion was surgically resected  Cavernoma, also known as cavernous malformation, cavernous angioma, or cavernous haemangioma, is the second most common cerebrovascular lesion, with a reported incidence of 0.5% , 2. Abouvia the right retrosigmoid approach. The lesion was dark-red, had an abundant blood supply, and contained vessel-like structures  showed a well-demarcated, oval mass measuring 17 × 11 × 10 mm in the right cerebellopontine angle . The lesructures . In ordeructures . Sensoryructures . Followith postoperative day. There was no recurrence of pain during the 3-year follow-up period. Her postoperative facial hypoesthesia on the right side was not improved.The pathological diagnosis was cavernoma. Microscopically, malformed vascular tissue and hyaline degeneration of the vascular wall were observed . PostopeTN is the most common form of craniofacial neuropathic pain, which can be classified into 3 types, idiopathic, classic, and secondary TN . Idiopatvia craniotomy is the primary management strategy, and most patients have good outcomes after surgery  for the publication of any potentially identifiable images or data included in this article.HL, CC, and YL collected the data and prepared the manuscript. JL analyzed the data and created the tables and figures. XY and LC designed and supervised the work. All authors agree to be accountable for the content of the work, contributed to the article, and approved the submitted version.This work was supported by National Natural Science Foundation of China, Grant/Award Number: 82172680.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.https://www.frontiersin.org/articles/10.3389/fneur.2022.982503/full#supplementary-materialThe Supplementary Material for this article can be found online at: Click here for additional data file.


Jejunal divertica is a rare entity with an often clinically silent course. However, it may be associated to life-threatening complications such as perforation. Therefore it should be considered in every case of acute abdomen.A 60-year-old female presented with a generalized abdominal pain associated with vomiting evolving for 24 h. Physical examination found an irreducible and tender hernia in the umbilical region with abdominal guarding. Laboratory test results showed a biological inflammatory syndrome. The primary diagnosis of strangulated umbilical hernia was suspected and the patient underwent an emergency laparotomy. Intra-operative examination revealed mutiple jejunal diverticula, with a perforation in one diverticulum leading to generalized peritonitis. A bowel resection and peritoneal lavage were performed with good outcome.Jejunal diverticula is a challenging condition with various non-specific clinical presentations. Jejunal perforation is its most feared complication. Deceitful abdominal examination among elderly patients and lack of specific signs may lead to diagnostic delay responsible for high mortality rate and poor prognosis. Adjunctive imaging modalities may be needed to help establish a prompt diagnosis and dictate management strategy. Treatment of perforated jejunal diverticulum is based on limited bowel resection associated to primary anastomosis.Jejunal diverticulitis should be kept in mind as a differential diagnosis in every case of acute abdomen. High index of clinical suspicion and eventual further radiological examinations are required to avoid misdaignosis and save patients' lives. •Isolated jejunal diverticula perforation is extremely rare condition.•Complicated jejunal diverticulitis is a diagnostic challenge with a high mortality rate.•Jejunal diverticula might be difficult to identify and is rarely encountered by young surgeons.•It is important to include this condition in the differential diagnosis of acute abdomen. She had no significant past family history and she denied any tobacco, alcohol or any drug use. The pain suddenly started 24 h before admission and gradually increased. Physical examination revealed a blood pressure at 110/80 mmHg, a body temperature at 38.5 °C, a respiratory Rate at 22/min, and a heart rate at 106 beats/min. An irreducible and tender hernia was found in the umbilical region with abdominal guarding. Laboratory test results showed high levels of white blood cells , high rate of C-reactive protein (100 mg/dL), and low levels of hemoglobin (10.6 g/dL). Whereas urea, creatinine, and lipase values were within the normal limits. The primary diagnosis of strangulated umbilical hernia was suspected and the patient underwent an emergency laparotomy, via median incision. The surgery was conducted by a senior surgeon with ten years surgical specialty experience. Afterward, a fibrinous exudate was noticed in the abdominal cavity. Exploration of the bowels . One jej3Excluding Meckel's diverticulum, small bowel diverticula are acquired herniations of the mucosa and submucosa through weakened areas of the muscularis layer The most common part of the small bowel to be affected by diverticula is the proximal jejunum (75%), followed by the distal (20%) and then the ileum (5%) Jejunal diverticulum etiology has not been definitively identified. However, intestinal dysmotility which leads to abnormally elevated intraluminal pressure, and the structural weakness of penetration areas of the vasa recta blood vessels and nerves have been thought to play a role Most patients with jejunal diverticula are asymptomatic and in the majority of cases, jejunal diverticulosis is diagnosed incidentally either on imaging or intraoperatively Perforation of the jejunal diverticula is a serious complication that occurs in 2 to 6% of cases Diagnosis is challenging since there are no pathognomic signs or symptoms. Besides, elderly individuals may present a challenge in diagnosis due to the lack of clear physical signs, which explains the need for further examinations According to the literature reporting 290 cases of jejunal diverticula 4Given its low incidence, complicated jejunal diverticulitis can be both a diagnostic and therapeutic challenge with a high mortality rate. Accordingly, although jejunal diverticulitis is rare, it is important to include it in the differential diagnosis of patients with abdominal pain. Indeed, early recognition and prompt surgical intervention are critical to improve patients' outcome.CTcomputed tomographyMRImagnetic resonance imagingNone.An ethical approval was obtained from Jendouba Regional Hospital Medical Ethics Committee N° CC04Y22. We confirm that all methods were performed in accordance with the ethical guidelines of the 1975 Declaration of Helsinki.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Conceptualization: AM.Data curation: KA.Supervision and performing surgery: HZ.Writing - original draft:MH.Writing - review & editing: AM.The final version of manuscript was read and approved by all authors.N/a.Atef Mejri.Not commissioned, externally peer-reviewed.The authors declare that they have no conflicts of interest.