Bronchial schwannomas are extremely rare among the benign tracheobronchial tumors and little are known about its epidemiology and optimal clinical management. Here, we report a case of bronchial schwannoma in a young Japanese man and clinical implications about epidemiology, symptom, diagnosis, and treatment of bronchial schwannoma.A 37-year-old man visited our department with a nodule incidentally found on his chest radiograph during a routine medical checkup.The tumor was diagnosed as a bronchial schwannoma after pathological evaluation. Microscopically, the tumor consisted of spindle cell proliferation characterized by an alternating highly ordered cellular Antoni A component with occasional nuclear palisading and a loose myxoid Antoni B component. Tumor cells were immunoreactive for S100 but not for smooth muscle actin or KIT.A video-assisted right middle and lower bilobectomy was performed.He remains under observation without recurrence.In our review, many reports have come from Asian countries. Bronchial schwannoma can occur within a wide range of age groups and in both men and women. No difference in incidence was observed between right and left bronchial tree. Bronchial schwannoma is sometimes difficult to differentiate from malignant diseases. We should include bronchial schwannoma as one of the differential diagnoses of primary bronchial tumors. In addition to presentation of our case, we reviewed published cases of bronchial schwannomas, evaluated their epidemiology, and tried to clarify these issues.Schwannoma is a benign encapsulated nerve sheath tumor arising in peripheral nerves. Bronchial schwannomas are extremely rare and account for 2.2% of benign tracheobronchial tumors.A 37-year-old Japanese man visited our hospital after a nodule was found on chest radiograph during a routine medical checkup. He was asymptomatic and had no past medical history and no family history of malignant disease within the second degree of kinship. He was an office worker with no smoking or dust exposure history.Vital signs, including body temperature and oxygen saturation, were normal. No wheeze or crackle was heard on auscultation. No lymph nodes were palpable.Milestones related to diagnosis and intervention is shown in Table A video-assisted right middle and lower bilobectomy was performed. Gross examination of the nodule showed a dumbbell-shaped solid tumor surrounded by a fibrous capsule with a mottled yellow-white appearance on the cut section. A loupe view is shown in Figure The patient had a good postoperative course and remains under observation in an outpatient clinic. Image findings show that the tumor has not recurred to date.–28 The process of selecting the cases is shown in Figure We searched the cases with the keyword “bronchial schwannoma” via PubMed in October 2021 and identified 112 articles. We identified 33 cases of bronchial schwannoma among them (Table ,29 However, cases have been reported in which bronchial schwannomas were found incidentally through bronchoscopy in patients without symptoms. Moreover, some cases are discovered on chest radiographs during medical checkups, such as this case. Although FDG-PET/CT is helpful in distinguishing between benign and malignant lung tumors, maximum standardized uptake values on FDG-PET/CT of schwannomas has been shown to be varied, probably due to the degrees of cellularity, microvascular density or vascular permeability.,31 It should be noted that FDG-PET/CT is not always practical in differentiating a schwannoma from a malignant tumor, as a schwannoma sometimes shows abnormal FDG accumulation, as was the case in this patient. Histological evaluation is the standard method used for the diagnosis of schwannoma. The presence of typical Antoni A and Antoni B patterns and expression of S100 protein helps the pathological diagnosis of schwannoma.,18,29 Surgical resection or resection by less-invasive techniques is the usual treatment for bronchial schwannomas. Resection with bronchoscopy has been attempted instead of surgical resection.,32 The tumor should be resected completely to avoid local recurrence due to incomplete resection. In this case, we could not confirm the diagnosis before surgical resection as the tumor bled easily and therefore could not be biopsied through bronchoscopy. The results from FDG-PET/CT could not rule out a malignant tumor. Without resection, the growing tumor was expected to cause obstructive pneumonia as prior radiographs had not been reported to demonstrate a nodule.Bronchial schwannomas are often detected and diagnosed with clinical presentations of cough, hemoptysis, and dyspnea; and some cases are diagnosed after the development of obstructive pneumonia.In conclusion, bronchial schwannoma is a rare tumor but should be included as one of the differential diagnoses of primary bronchial tumors. The epidemiology of bronchial schwannomas is not fully understood, but they can occur within a wide range of ages and in both men and women. Accurate examination, diagnosis, and treatment should be made correctly. Further investigation and accumulation of cases is needed.The patient achieved histopathological confident diagnosis of the tumor and to rule out the possibility that it might be malignant tumor. The accurate diagnosis and successful treatment relieved the patient of developing additional difficult comorbidities including obstructive pneumonia or lobar atelectasis.Conceptualization: Yosuke Aoyama, Atsushi Miyamoto, Takeshi Fujii, Sakashi Fujimori, Meiyo Tamaoka, Daiya Takai.Data curation: Yosuke Aoyama, Takeshi Fujii.Formal analysis: Yosuke Aoyama, Takeshi Fujii.Investigation: Yosuke Aoyama, Atsushi Miyamoto, Takeshi Fujii, Sakashi Fujimori.Methodology: Yosuke Aoyama, Atsushi Miyamoto, Sakashi Fujimori, Meiyo Tamaoka, Daiya Takai.Project administration: Atsushi Miyamoto, Takeshi Fujii, Sakashi Fujimori, Meiyo Tamaoka, Daiya Takai.Resources: Sakashi Fujimori.Supervision: Atsushi Miyamoto, Takeshi Fujii, Sakashi Fujimori, Meiyo Tamaoka, Daiya Takai.Validation: Yosuke Aoyama, Takeshi Fujii.Visualization: Yosuke Aoyama, Takeshi Fujii.Writing – original draft: Yosuke Aoyama.Writing – review & editing: Yosuke Aoyama, Atsushi Miyamoto, Takeshi Fujii, Sakashi Fujimori, Meiyo Tamaoka, Daiya Takai.


Teaching Point: Although frequent, the coexistence of diffuse hepatic hemangiomatosis (DHH) with giant cavernous hemangioma (GCH) should be mentioned clearly and communicated to the referring physician for an appropriate management. Figure 1a, b). Dynamic gadolinium-enhanced images in the axial plane showed peripheral nodular enhancement in the arterial phase and partial centripetal filling in the delayed phases, which is characteristic of GCH . Significant mass effect on adjacent organs and vasculature was observed, However, there were no signs of rupture or hemorrhage. In addition, an ill-defined area of confluent hyperintense nodules separated by hypointense septations was seen in posterior segments adjacent to GCH. This area showed heterogeneous discontinuous nodular enhancement in arterial phase which gradually increased in delayed phase . The left lobe has expanded without any lesion. The diagnosis of GCH with adjacent DHH was made and the patient was referred to surgical department for therapeutic management.A 56-years-old female with no past medical history presented with suspected atypical hemangioma of the liver causing chronic abdominal discomfort. Laboratory tests were normal. At Magnetic resonance imaging (MRI), axial and coronal single-shot fast spin-echo T2-weighted images revealed a 19 × 18 × 16 cm well-defined mass with lobulated margins replacing the right liver. The mass was hyperintense with central loculations of higher signal intensity and numerous thin hypointense septa (Giant cavernous hemangioma (GCH) is an atypical form of hepatic hemangioma characterized by a large size, exceeding 5 cm. Symptomatic lesions require surgical management, which depends not only on the tumor’s size but also on the quality of residual liver parenchyma . DiffuseUnlike GHC, which have smooth well-defined margins with classic findings on imaging modalities, DHH appears as an ill-defined area often adjacent to GCH, forming by scattered or confluent innumerable small nodules hypodense on unenhanced CT, hypointense on T1-weighted and hyperintense on T2-weighted images [Management strategies of symptomatic GCH depend on the presence and extent of DHH. These cases can be difficult to enucleate and are better managed by extensive lobectomy or hepatic transplantation. Thus, imaging assessment of residual hepatic parenchyma is highly recommended .


Congenital hand anomalies are common, and must always be considered as a differential diagnosis in patients with hand pathology. We report the case of a child who sustained a fingertip injury to highlight an unusual presentation of central polydactyly. The child had not sustained any other injuries and was otherwise healthy. He had no significant past medical history or family history of any conditions. He was right‐hand dominant, lived with his mother and father at home, and attended primary school. Clinical examination of the left middle finger revealed an ulnar‐sided subungual hematoma, and an erythematous and tender fingertip Figure . ApproxiThe patient underwent a nailbed exploration under general anesthetic, and the operation was performed by a consultant plastic hand surgeon: the nail plate was removed, and while no nailbed lacerations were identified, there was a small central nailbed sulcus Figure . Assessm3Considering the radiological appearance of our patient's left middle finger and the observable nailbed sulcus between the two bone ‘horns’ of the bifid distal phalanx, we believe to have identified a subtle, asymptomatic central polydactyly, brought to our attention only by the coincidental traumatic injury. As far as the authors are aware, this is a unique presentation for the diagnosis of this extremely rare congenital anomaly.While tuft fractures are common injuries following trauma to the fingertips,Surgery for central polydactyly has been regarded as difficult, especially when complicated by co‐existing syndactyly, and patients require individualized treatment to create a functional and aesthetic hand.4Injuries to the hand are common, but it is critical for clinicians to assess all patients individually to ensure accurate diagnosis. We report this case to highlight the unusual presentation of a rare congenital hand anomaly masquerading as a common traumatic injury, and the initial diagnostic uncertainty we faced. Methodical clinical examination combined with careful interpretation of the radiological images were essential in determining the correct diagnosis, and therefore planning appropriate management. We recommend that clinicians consider the possibility of a congenital bifid distal phalanx in patients who have sustained fingertip trauma and there appears to be a longitudinal split fracture, but no soft tissue damage is present.Mahir Bhoora: Conceptualization; data curation; formal analysis; investigation; methodology; project administration; visualization; writing – original draft; writing – review and editing. Dallan Dargan: Conceptualization; investigation; writing – review and editing. Chris Milner: Conceptualization; investigation; supervision; validation; writing – review and editing.The authors received no financial support for the research, authorship, and/or publication of this article.The authors declare no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.Given the case report nature of this publication, ethical approval from a local ethics committee was not required. All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation  and with the Declaration of Helsinki 1975, as revised in 2008.Written informed consent was obtained from the patient's parent.


Intramuscular myxoma (IM) is a rare benign tumour. It may occur at any age but most commonly occurs among older women. The preoperative diagnosis of IM is complicated. The diagnosis can only be definitively established by histopathological examination. The treatment of choice is radical surgical excision. We report a case of a 41-year-old woman with an IM of the biceps brachii muscle. Intramuscular myxoma (IM) is a rare, benign soft tissue tumour . UsuallyIn line with the SCARE  criteria, we report a case of a 41-year-old woman with an IM of the biceps brachii muscle .A 41-year-old female presented with a 3-year history of a slowly growing lump on the anterior side of the left upper arm. Her past medical history was significant for type 2 diabetes mellitus, diagnosed ~10 years prior to the presentation. Otherwise, the medical history was unremarkable.During physical examination, a 2 cm hard, non-tender lump was found in the distal part of the left biceps brachii muscle. The examination revealed no motor or sensory deficits distal to the site of the lesion, and there were no restrictions in the movement of the arm as compared with the unaffected side. Blood laboratory findings were within normal limits.An ultrasound scan revealed a 2.0 × 2.0 cm well-defined heteroechogenic lesion with a cystic component in the left biceps brachii muscle .The patient underwent local with clear margins surgical excision of the lesion. During surgery, the lesion appeared as a solid, well-circumscribed, encapsulated mass, and it was easily removed .Histopathological examination of the surgical material revealed the lesion to be an IM. The tumour was surrounded by muscle fibres and composed of spindle- and stellate-shaped cells with small, hyperchromatic nuclei  and 4.The patient’s postoperative course was uneventful. She did not require any adjuvant treatment. There was no recurrence after 1 year of follow-up.The IM is a rare tumour with an incidence of approximately one per million . It may GNAS  gene [The aetiology of the IM is still unknown , althougng) gene , 10. Theng) gene  or very ng) gene . Single ng) gene , and it It is difficult to obtain a definitive IM diagnosis through non-invasive diagnostic methods, as the tumour presents itself with non-specific clinical symptoms and is easily mistaken for other tumours in imaging studies.Clinically, an IM most frequently manifests itself as a slow-growing, palpable, painless mass , as was Imaging techniques may include X-ray, ultrasound, computed tomography (CT scan) or magnetic resonance imaging (MRI). X-ray examinations may be normal or show a non-specific intramuscular mass that rarely contains calcifications , 12. On The tumour should be differentiated from other benign lesions  and malignant tumours  , 12, 13.Although imaging techniques are essential in the diagnostic workup, a definitive IM diagnosis can only be reached through histopathological examination. Core needle biopsy might be useful for differential diagnosis prior to treatment.Macroscopically, IMs typically appear as oval, grey-white, gelatinous masses with a firm consistency , 3. MicrThe treatment of choice for IM is radical surgical excision. Complete excision of the lesion with clear small margins of surrounding tissue is essential whenever feasible . IncomplIMs do not show potential for malignant transformation and do not have a tendency to metastasize .None declared.None.


Ganglioneuroblastomas (GNBs) are a rare subtype of neoplastic tumors that arise from the autonomic nervous system and contain both mature gangliocytes and immature neuroblasts. The primary age group affected by GNBs is the pediatric population, with less than 50 cases of adult GNBs existing in the literature. To the authors’ best knowledge, only 21 cases of GNBs arising in the adrenal glands of adults have been reported. Herein we present a literature review examining the symptoms, treatment type, age, and sex of adults, and the presence of tumor metastases and calcification from the 21 cases reported in the literature. Neuroblastic tumors arise from primitive cells of the sympathetic nervous system and are composed of three histological subtypes: neuroblastoma, ganglioneuroblastoma (GNB), and ganglioneuroma ,2. TheseGNBs contain both mature gangliocytes and immature neuroblasts and are composed of two subgroups, nodular-GNBs (stroma poor) and intermixed-GNBs (stroma rich) . The majThe inclusion criteria for our study consisted of all cases in the literature that reported an adrenal GNB in an adult (>18 years of age). Searching PubMed and Google Scholar for articles of this type yielded 21 cases. A literature review, examining the symptoms, treatment type, age and sex of the patients, and presence of tumor metastases and calcification from these 21 cases, was conducted , ultrasound, and magnetic resonance imaging (MRI), are of great importance when trying to diagnose GNBs. These imaging modalities can help characterize the mass and are useful in pretreatment risk stratification ,30. CT fRegardless of age or tumor location, it is important to keep examining the patient periodically after treatment of GNB, due to the chance of recurrence which mostly occurs within the first two years after surgery ,31. ExamGNBs are a rare subtype of neoplastic tumors that contain both mature gangliocytes and immature neuroblasts. Less than 50 cases of adult GNBs exist in the literature, with only 21 cases of GNBs arising in the adrenal glands of adults having been reported. A literature review, examining the symptoms, treatment type, age, and sex of the patients from the 21 cases of adult-onset adrenal GNB in the literature was conducted. From the review, it was concluded that adrenal GNBs of adult-onset affect males at a slightly higher rate than females, but not to the magnitude that is reported in the current literature by previous literature reviews on this topic, are diagnosed more in the 20-29-year-old patient population than any other adult population, can present with a varying degree of symptoms, are mostly treated by surgical removal of the tumor, and sometimes express metastatic characteristics and calcification.


TP53 mutations have been frequently reported in patients with Li–Fraumeni syndrome (LFS), resulting in a predisposition to various malignancies. Mutations other than germline TP53 mutations can also cause LFS-associated malignancies, but their details remain unclear. We describe a novel c-myc amplification in a unique liposarcoma in a patient with LFS.Germline HER2 amplifications. Computed tomography revealed an anterior mediastinal mass, which was surgically resected. Histological analysis revealed three different lesions corresponding to myxoid liposarcoma-, pleomorphic liposarcoma-, and well-differentiated liposarcoma-like lesions. Fluorescence in-situ hybridization (FISH) analysis did not detect MDM2 amplification, Rb1 deletion, break apart signals of EWS, FUS, DDIT3, or c-myc, or c-myc-IGH fusion signals, but it did detect more c-myc signals. Further FISH analysis and comprehensive genomic profiling revealed c-myc amplification. We considered two differential diagnoses, dedifferentiated liposarcoma lacking MDM2 amplification and myxoid pleomorphic liposarcoma (MPLPS), and determined that this case is most likely MPLPS. However, definite diagnosis could not be made because a clear-cut differentiation of the case from liposarcomas was not possible.A female patient with LFS developed breast cancer twice at the age of thirty; both were invasive ductal carcinomas harboring c-myc amplification could not be detected in various liposarcomas, but the present unique liposarcoma showed c-myc amplification, so the c-myc amplification may indicate that the present liposarcoma is an LFS-related tumor. The present case further clarifies the pathological features of MPLPS and LFS-related liposarcomas by broadening their histopathological and genetic diversities.A previous study demonstrated that  TP53 mutations have been frequently reported in patients with Li–Fraumeni syndrome (LFS), resulting in a predisposition to various malignancies [TP53 can also cause LFS-associated malignancies, but their details have not been reported. The most common tumor arising in TP 53 mutation carriers is breast cancer, followed by soft tissue sarcomas, brain tumors, osteosarcomas, and adrenocortical tumors [Germline gnancies . Mutatiol tumors –4. AmongMDM2 amplification in WLPS and DLPS, fusion of EWS, FUS, and DDIT3 in MLPS, and Rb1 deletion in PLPS, has led to the development of subtype-tailored management and novel systemic therapies [RB1 expression [MDM2 amplification and fusion of EWS, FUS, and DDIT3 [Liposarcomas (LPSs) are sarcomas of adipocytic differentiation. These tumors are pathologically classified into five subtypes: well-differentiated LPS (WLPS), dedifferentiated LPS (DLPS), myxoid LPS (MLPS), pleomorphic LPS (PLPS), and myxoid pleomorphic liposarcoma (MPLPS) . Improveherapies . MPLPS ipression , and it nd DDIT3 . Furthernd DDIT3 ; howeverc-myc amplification in a unique MPLPS-like liposarcoma in a female patient with LFS using histological, immunohistochemical, and fluorescence in-situ hybridization (FISH) analysis and comprehensive genomic profiling (CGP).Here, we describe a novel TP53 mutations [HER2 amplifications identified by FISH analysis, with no metastasis to axillar lymph nodes. No clinically significant mutation was identified by BRCA1 and BRCA2 analysis a the blood sample. The patient’s family history revealed that her mother developed left and right breast cancers, a brain tumor, and lymphoma before the age of 45, so that the patient clinically fulfilled the Clinical Criteria for Classic Li-Fraumeni syndrome [A female patient with LFS, diagnosed by DNA sequencing of a blood sample in which heterozygous pathogenic utations , c.818G syndrome .About two weeks after breast lesions had been identified, computed tomography also revealed an anterior mediastinal mass Fig. A, which Immunohistochemically, p53 Fig. H and c-mMDM2 amplification ; MYC amplification observed (absolute copy number: 59); Rb1 Splicing Variant c.1422-1G>T and TP53 c.818G>A observed. As variants of unknown significance or benign variants, BRAF c.64G>A; LTK c638 639ins TGGCGG; MST1R c.679G>T; NOTCH3 c.4039G>C; TSC2 c.2409G>T; SOX9 amplification (absolute copy number: 69); MYC rearrangement exon 2; NOTCH3 rearrangement exon 1; and SOX9 rearrangement exon 3 were detected. For recurrent liposarcoma, chemotherapy with gemcitabine and docetaxel was administered. However, pleural effusion appeared during this treatment, rendering it ineffective. Pleural effusion cytology detected malignant cells, suggesting liposarcoma  MPLPS. As a DLPS, the PLPS-like area may correspond to homologous lipoblastic differentiation [MDM2 [RB1 expression [Rb1 deletion was not detected; thus, the Rb1 splicing variant c.1422-1G>T detected by CGP was supposed to be associated with Rb1 inactivation. The lack of MDM2 amplification and fusion of EWS, FUS, and DDIT3 was consistent with MPLPS [Histologically, the tumor consisted of heterogeneous components; MLPS-like, PLPS-like, and WLPS-like areas. Together with the abovementioned molecular findings, we considered two possible differential diagnoses: 1) DLPS lacking ntiation , althougon [MDM2 , 11, buton [MDM2 . Collecton [MDM2  and assoon [MDM2 . Moreoveon [MDM2 , showingpression . In the th MPLPS . To the th MPLPS , 16. Thec-myc amplification in the present tumor, in addition to germline TP53 mutations. C-myc is frequently dysregulated in various neoplasms, and its amplification has also been reported in the tumor from patients with LFS [TP53 mutation was associated with various oncogene amplifications [c-myc amplification could not be detected in various liposarcomas in a past study [c-myc amplification, so the c-myc amplification may indicate that the present liposarcoma is an LFS-related tumor. Furthermore, in various sarcomas, including liposarcomas, c-myc overexpression was reported to be correlated with poor prognosis [We detected a with LFS ; its ovewith LFS , 18. Preications , 20. In st study , but therognosis –23, and c-myc amplification in a unique liposarcoma in an LFS patient. The present case further clarifies the pathological features of MPLPS and LFS-related liposarcomas by broadening their histopathological and genetic diversities.In summary, we identified a novel


In this paper, we present the case of acute airway obstruction due to tracheal carcinoma in a patient with glottic stenosis due to previously treated laryngeal carcinoma. Because of severe dyspnea from the obstructive endotracheal mass, tracheotomy under local anesthesia was immediately performed. Intubation with pediatric size (I.D. 4.5 mm) cuffed endotracheal tube was performed by the surgeon through tracheostomy under endoscopic visualization. Blakesley forceps and electrocautery were used for tumor debulking. Postoperatively there were no complications and the patient was discharged after four days. The histopathology report showed a squamous cell carcinoma. The tumor board decided on adjuvant chemoradiotherapy for the treatment of the patient. Primary malignant neoplasms of the trachea are very rare. With an incidence of 0.1 per 100,000 persons per year, these tumors account for 0.2% of respiratory tract malignancies and approximately 0.02-0.04% of all malignancies ,4. SCC i2. Five years ago, he was diagnosed with a T2 glottic SCC of the larynx and treated with transoral laser cordectomy, a temporary tracheostomy, and adjuvant radiotherapy  elsewhere. Examination with a flexible endoscope showed severe stenosis of the glottis  due to postoperative adhesions, especially in the anterior commissure, and an endotracheal tumor with occlusion of about 85% of the trachea lumen. There was no evidence of malignancy in the larynx. Also, there were no palpable lymph nodes in the neck. The patient had a recent computed tomography (CT) scan that showed a large obstructive endotracheal mass arising from the posterior tracheal wall with dimensions of 2.4 x 1.4 x 4.5 cm, approximately, with no evidence of lymph node metastases or locoregional invasion  and his BMI was 20 kg/mn Figure , 1b.The tumor distance from the glottis was approximately 4 cm, and the distance from the carina was approximately 4.5 cm. Because of severe dyspnea and low blood oxygen saturation, we immediately performed a tracheotomy under local anesthesia between the second and third tracheal rings (at the previous scar). Using a 45° rigid endoscope that was inserted from the tracheostomy, we managed to visualize all the tumors, which extended below the tracheostomy until a few cm above the carina Figure , 2b, 2c.Then, under endoscopic visualization, intubation was performed with a pediatric-size (I.D. 4.5 mm) cuffed endotracheal tube from the surgeon through tracheostomy to avoid injury to the tumor and bleeding. General anesthesia was administered by the anesthesiologist. Debulking was performed with Blakesley forceps and bipolar electrocautery. Suction was used constantly to prevent blood from entering the lung and bronchi. Specimens were sent for histopathological examination. At the end of the operation, we placed an extended-length cuffed tracheostomy tube with an interior diameter of 8 mm. Postoperatively, there was no need for an intensive care unit. There wasn’t any complication or need for oxygen therapy. A postoperative endoscopic examination was performed Figure , and theThe biopsy revealed an SCC with moderate-to-low differentiation. The tumor board decided on postoperative chemotherapy and radiation therapy for the patient after consultation. The patient decided to undergo chemoradiotherapy in another center and received a dose of 64 Gy of radiotherapy with concurrent chemotherapy. One year after surgery, the patient was alive with good local control of the disease but unfortunately presented with brain metastasis.Primary tracheal carcinoma is the rarest malignancy of the airway, and this can be attributed to the role of local mucosa in immunosurveillance . SCC is For radiological evaluation, we used the CT of the neck and chest that the patient brought when he presented to the emergency department. CT is the most appropriate exam that provides information about the extension of the tumor, the depth of invasion, the invasion of adjacent structures, and the presence of lymphogenic or distant metastases. It can also provide excellent spatial resolution and has the ability to generate three-dimensional reconstructions, which allows for rapid evaluation of the extent of tracheal pathologies. Magnetic resonance imaging (MRI) is helpful in imaging tracheal compression or invasion by mediastinal masses or vascular anomalies and fluorodeoxyglucose positron emission tomography (FDG PET)/CT is reserved for staging patients with tracheobronchial malignancies. In our case, the carina was not involved, and there was no evidence of lymph node metastasis or invasion of adjacent organs. According to the CT, the extension was 4.5-5 cm in length, and more than 85% of the tracheal lumen was obstructed. In order to perform surgery with the goal of free margins of resection, it would be necessary to resect a large part of the trachea, including the previous tracheostomy scar, and in this case, an end-to-end anastomosis would present difficulty and excessive tension. In a previously irradiated trachea, this could lead to severe, life-threatening postoperative complications.Debulking with interventional rigid bronchoscopy could be very risky and even fatal in our case. Endotracheal intubation in a patient with severe glottic stenosis and a high degree of endotracheal obstruction who already had severe dyspnea could be impossible and even dangerous, causing complete airway obstruction. Unsuccessful oral endotracheal intubation attempts might cause severe bleeding from the tumor, which wouldn’t be easy to control, leading to airway compromise and death. Hence, to manage the airway safely, we chose to perform a tracheotomy under local anesthesia and then proceed with intubation through it under direct endoscopic visualization. In this way, with the lung and bronchi protected, we performed debulking and managed to prevent airway obstruction and thus save the patient’s life.Tracheal tumors are rare malignancies and management of acute airway obstruction in these cases can be very challenging. CT is necessary for the diagnostic procedure and surgical resection is advocated as the procedure of choice for complete excision when it is possible. In this paper, we document a case of acute airway obstruction from a large endotracheal tumor which was not feasible to be totally resected. In cases like this, the aim is to save the patient’s life which can be achieved by performing tracheotomy and tumor debulking from the tracheostomy. After that, the patient could have the opportunity to undergo chemoradiotherapy and also have the chance of better overall survival.


Several studies have previously documented the development of complications stemming from injection with one of the various COVID-19 vaccines. No study, however, has discussed the spontaneous development of a soft tissue mass shortly after a COVID-19 vaccine injection. We report on 66-year-old female with concerns of a growing shoulder mass, 2 weeks after receiving a COVID-19 vaccine booster. Initial work-up with X-ray and MRI was concerning for a soft tissue neoplasm, specifically a soft tissue sarcoma. Subsequent ultrasound guided biopsy demonstrated a benign granulomatous lesion. No further management was required as the lesion spontaneously resolved during a 3-month follow-up period. It has been previously documented that vaccine components can produce granulomatous inflammation. One study reported postvaccination granulomas in children following the pertussis immunization A 66-year-old female presents to our orthopedic clinic with concerns of a right shoulder rapidly expanding palpable mass. This female with a medical history of rheumatoid arthritis (RA) and rotator cuff surgery presents to our outpatient orthopedic sports medicine clinic concerned about a mildly tender and rapidly enlarging right shoulder soft tissue mass of three weeks duration. She denies systemic symptoms of fever, appetite changes, or weight loss, as well as any antecedent major trauma or other injury. The patient had received the COVID-19 Moderna vaccine booster (3rd dose) in the same deltoid region 4 weeks prior. She notated the injection had been associated with more pain than the prior doses.On physical exam, a nonmobile firm mass was palpable along the superolateral margin of the right deltoid muscle, without overlying skin changes. Neurovascular and motor testing were negative for weakness and sensory deficits. Shoulder radiographs revealed a soft tissue mass in the region of the lateral deltoid, without calcification or underlying osseous involvement . SubsequWe present a case of a 66-year-old woman who developed a localized granulomatous reaction that presented as a mass following a COVID-19 Moderna Booster vaccine Several case reports have documented the development of granulomatous inflammation following vaccine administration. One study reported postvaccination granulomas in children following the pertussis immunization. This same group identified that the ingredient responsible was an aluminum salt adjuvant as 77% of children who developed granulomatous inflammation also developed a contact allergy to aluminum We hereby confirm consent was granted for the publication of this case by the patient, as so long as all records are de-identified.


Tumors of the uterine tube are rare pathologies representing less than 1 % of all gynecologic cancers; they are dominated by adenocarcinomas. Secondary metastatic forms are the most frequent, whereas primary tumors are very rare and represent only 10 %, which suggests that the fallopian tube is an organ with low oncogenic potential.We report the case of a patient followed in the gynecology department C of the CHU IBN ROCHD CASA for a primary tubal adenocarcinoma, with a history of breast cancer.The diagnosis of its origin is difficult preoperatively, the treatment and staging are the same as for ovarian cancer.The treatment is also identical to the management of ovarian cancer, but their prognosis is better because they are most often diagnosed at an earlier stage. All our work has been reported in line with the SCARE criteria and guidelines 2This is a 62-year-old female patient, IGIP, postmenopausal, never had oral contraception, no notion of recurrent genital infection, She had undergone subtotal hysterectomy with bilateral adnexectomy in 2015 for a 10 cm right latero-uterine mass. The anatomopathological study showed a right intra tubal serous adenocarcinoma, grade III, without ovarian involvement or invasion of the epiplon. The patient received 6 courses of chemotherapy, the CA 125 was 192, a trachelectomy was performed 1 year later with non-specific fibrous remodeling with chronic exocervicitis, without signs of malignancy. 4 years later the patient consulted for chronic pelvic pain for 6 months, of increasing intensity, without sign of urinary or digestive compression. The clinical examination revealed a patient in good general condition, with good vitals, a supple abdomen, no clinically palpable mass, the vaginal slice was clean without lesions, the senological examination and the rest of the somatic examination were without particularity.A pelvic MRI was performed showing a solid cystic mass measuring 52 mm in width, 42 mm in anteroposterior diameter, 64 mm in height, in contact with the bowel and the bladder without any sign of infiltration and without associated adenopathy , CA 125:The exploration revealed a mass adherent to the right iliac vein without invading it, an excision was performed with simple postoperative follow-up with anatomopathological examination: tubal adenocarcinoma grade III, then the patient was referred to adjuvant chemotherapy.3Tubal cancer is the rarest cancer of the genital tract representing 0.1 to 1.98 % of all gynecologic cancers The symptoms are not specific, often associating a pelvic mass, bleeding, and pain, but the pathognomonic triad is hydropstubaeprofluens associating abundant fluid loss, pelvic colic, and a mass is described in only 10 to 15 % of cases The first paraclinical examination to be requested is ultrasonography, which often shows the same aspect of epithelial carcinomas of the ovary in the form of a mixed adnexal mass, solid and cystic, hypervascularized on Doppler Staging is based on ovarian cancer The treatment is also identical to the management of ovarian cancer, but their prognosis is better because they are most often diagnosed at an earlier stage The prognosis depends on tumor size The 5-year overall survival of tubal malignancies is estimated to be 44 % according to major series 4Tubal adenocarcinoma is a rare entity, of unknown etiology, underestimatedor often confused with ovarian pathology with which it shares the same treatment and staging, the positive diagnosis is difficult because the clinical picture is polymorphic, MRI brings a great diagnostic interest, the prognosis depends on the FIGO stage.Not commissioned, externally peer-reviewed.None.I declare on my honor that the ethical approval has been exempted by my establishment.Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient.NABILA YOUSSOUF: Corresponding author writing the paperSAMI ZINEB: writing the paperWATIK.FEDOUA: writing the paperSABIR SOUKAINA: writing the paperBOUFETTAL Houssin: correction of the paperMAHDAOUI Sakher: correction of the paperSAMOUH NAIMA: correction of the paperresearchregistry2464.DR YOUSSOUF NABILA.The authors declare having no conflicts of interest for this article.


Ruptured mesenteric hematoma is a rare entity that is not frequently reported in the literature. Spontaneous rupture of mesenteric hematoma has been vaguely described in a handful of cases. The majority of cases (unlike ours) had a reported etiology, like trauma, postoperative complications, connective tissue disease, and coagulopathy. To our knowledge, only two cases were reported for spontaneous rupture of idiopathic mesenteric hematoma. We, herein, present a 45-year-old male presented with abdominal pain, nausea, and vomiting associated with hypotension. His investigations, including abdominal computed tomography (CT) scan, showed a large lobulated mesenteric structure involving the small bowel mesentery, representing a mesenteric hematoma. Due to suspicion of intra-abdominal bleeding; we proceeded for exploratory laparotomy. Intraoperative findings showed a large mesenteric hematoma. The patient was stabilized after the evacuation of hematoma and no specific source of bleeding was identified. Ruptured idiopathic mesenteric hematoma is an extremely rare condition that poses a diagnostic challenge, it should be kept among deferential diagnoses, especially, in unstable patients with abdominal symptoms. Spontaneous mesenteric hematoma is a rare entity with vague etiology . It was We report a case of spontaneously ruptured idiopathic mesenteric hematoma in a 45-year-old male that was managed surgically at Hamad General Hospital, Doha, Qatar. A literature review in a comprehensive approach was carried out. To our knowledge, this is the third reported case in literature for spontaneous rupture of idiopathic mesenteric hematoma.3/µL , hemoglobin (Hb) 6.5 g/dL (13-17), platelets count 126 x103/µL (150-410), international normalized ratio (INR) 1.1, activated partial thromboplastin time (aPTT) 25.4 seconds (25-36), and lactic acid 3.5 mmol/L (<2.2).A 45-year-old male presented to the emergency department at Hamad General Hospital, Doha, Qatar, with abdominal pain for two days, associated with nausea, episodes of vomiting of food particles, and abdominal distension. He had no other gastrointestinal (GI) or genitourinary symptoms, no history of trauma or bleeding from other sites, and no history of anticoagulant or antiplatelets use. His past history was significant for type II diabetes mellitus (DM) and hypertension (HTN). The patient was a nonsmoker and nonalcoholic. Past surgical history was significant for left axillary abscess incision and drainage. On examination, the patient looked well and alert. His presenting vitals showed blood pressure (BP) of 91/58 mmHg, heart rate (HR) of 108 b/min, respiratory rate of 22, and temperature of 36.8 Celsius degree. On examining his abdomen, it was distended, with a palpable tender bulge in the left upper abdomen, and lower abdominal tenderness; however, there were no signs of diffuse peritonism. His labs showed white blood cell count of 12.5 x10Abdomen and chest x-rays were normal. Bedside ultrasound showed the presence of free fluid in the pelvis. Computed tomography (CT) scan of the abdomen showed a 15-cm structure originating from the small bowel mesentery, extending from the mesenteric root towards the right iliac fossa, likely representing a mesenteric hematoma and moderate amount of fluids in the abdomen, possibly hemoperitoneum. Another differential diagnosis on the CT scan was a soft tissue mass that ruptured and caused this bleeding Figures -1d.After initial resuscitation, the patient initially responded and had his CT scan. However, he started deteriorating again; hence, he was taken to the operating theater for exploratory laparotomy, intra-operative findings were hemoperitoneum (two liters of blood), mesenteric hematoma involving the mesentery of the small bowel extending to the root near superior mesenteric artery (SMA) origin, no actively bleeding vessel was identified. Evacuation of the hematoma was undertaken, and the abdomen was left with temporary closure (ABTHERA vac) for a second look later, no definite closure was undertaken as the patient was not stable and no clear bleeding source was identified or controlled.The patient was shifted to the intensive care unit (ICU) postoperatively. Second look laparotomy showed healthy bowel, serosal hematoma involving the hepatic flexure and mesenteric hematoma with no active bleeding, approximation of the rectus sheath was not possible; hence, the decision is made for ABTHERA vac dressing again, further diuresis to resolve bowel edema in order to allow approximation of the fascia, so planned for re-look laparotomy after 48 hours.The third look showed a dry site of previous hematoma evacuation, resolving hematoma of the mesocolon, no active bleeding and the bowel was healthy, closure of the abdomen with the posterior component separation technique was undertaken as it was not possible to bring the sheath in the midline despite all conservative measure including diuresis and muscle relaxation. On the same day, the patient developed hypotension (BP 74/50), tachycardia (HR 115 b/min), and a drop in Hb to 6 mg/dL (from 8.6 mg/dL), resuscitation was started, and he was shifted to the operating theater, where bleeding from the left inferior epigastric artery was identified, controlled and vac dressing was applied. Three days later, closure of the abdomen using anterior component separation was done. The patient then recovered gradually with the uneventful course, his wound was healing well, and he was discharged after 22 days of his initial presentation with stable vital signs and hemoglobin.Mesenteric hematoma in itself is a rare cause of intraperitoneal hemorrhage due to bleeding from peripheral mesenteric vessels . There aIdiopathic mesenteric hematoma is an extremely rare pathology that may have vague presentations . MajoritCT abdomen with contrast is the diagnostic modality of choice to reveal mesenteric hematoma . ImagingManagement of mesenteric hematoma is variable as it depends on the patient’s stability. If the patient is hemodynamically stable, he can be treated conservatively mainly with the absence of active bleeding . In clinOur patient presented with hypotension and features of hemodynamic instability. Due to the high suspicion of intra-abdominal catastrophe; bed side ultrasound demonstrated free fluid in the pelvis. Then an urgent CT scan was done after initial response to fluids. However, latter on his response to initial resuscitation was not sustained, the patient was explored via a midline laparotomy. Following abdominal closure; he was sent home in a good condition and a smooth postoperative course in six months follow up.Ruptured idiopathic mesenteric hematoma is an extremely rare clinical entity in patients with abdominal pain presenting to the emergency department. An abdominal CT scan with contrast is the diagnostic modality of choice to confirm the diagnosis as well as to rule out other potential causes of abdominal pain. Admission is warranted for all patients and the hemodynamic status of the patient guides the management. Bleeding can stop spontaneously or by therapeutic interventions. Thorough evaluation for the cause of bleeding is mandated in all cases to avoid recurrence.


Colonic diverticulosis is often asymptomatic, but it can complicate bleeding, abscess and stricture. Pneumaturia and faecaluria are unexpected manifestations of colonic diverticulitis complicated by colovesical fistula formation.This case report highlighted a 57-year-old man who presented with lower abdominal pain which was associated with pneumaturia and fecaluria. He was diagnosed with colovesical fistula after being misdiagnosed with other diseases from various clinic visits. Direct fluoroscopy was performed and a diverticular fistula of the sigmoid colon was confirmed by computed tomography of the abdomen. Hartmann's procedure and resection of the posterior wall of the bladder were resected.Pneumaturia and faecaluria are common but distinct manifestations of complicated diverticular diseases. Attention should be paid to general practitioners to achieve proper referral, hence early treatment and prevention of disease-related complications. •Colonic diverticulosis can be complicated by bleeding and stricture including fistula formation.•Pneumaturia and faecaluria are unexpected manifestations of colovesical fistula.•General practitioners should be aware of pneumaturia and faecaluria to reduce diverticulitis-related morbidity. It is cThe abnormal communication with the nearby structures will manifest a different presentation. When it involves the bladder, various clinical conditions will be manifested. Among those, patients can develop pneumaturia: a passage of gas in the urine forming bubbles; faecaluria: a passage of faeces in the urine; and recurrent urinary tract infection due to ascending infection. Besides diverticular fistula, other causes include diseases affecting the urinary system such as gas-forming urinary tract infection, emphysematous pyelonephritis, renal tumours or urinary catheterization. It is a challenging problem for general practitioners as these alarming presentations can be the presentations of many other disorders. We describe a 57-year-old man who presented with lower abdominal pain, pneumaturia and fecaluria in which a diagnosis of a colovesical fistula was made. This work has been reported in line with the SCARE criteria .2A 57-year-old man presented to the surgical outpatient department with a complaint of air bubbles and faecal matter in the urine. He had been suffering from lower abdominal discomfort for the last two years with occasional dysenteric symptoms. He went to a general practitioner and was treated with antibiotics for the gastrointestinal symptoms which gave him only temporary relief. He continued his work abroad with some available medications but the symptoms had worsened. He took a consultation with another doctor who prescribed steroids for the suspicion of ulcerative colitis. Five months after the start of symptoms, he developed dysuria and then he was treated for urinary tract infection with benign prostate hyperplasia. He still noticed the pneumaturia and fecaluria despite the medications. He was once referred to a psychiatrist for his bizarre symptoms. After 6 months, he was only advised to take an abdominal CT scan and colonoscopy.Direct fluoroscopy was performed and a diverticular fistula of the sigmoid colon was diagnosed. Colonoscopy revealed no malignant involvement. The condition was then confirmed by computed tomography (CT) of the abdomen. A laparotomy was performed and a fistula was visualized between the bladder and sigmoid colon A-C. Hart3Any medical practitioner undoubtedly must have gone through the complex cases thoroughly before coming to a final diagnosis especially if the symptoms persist. The possibility of extrapulmonary tuberculosis must be ruled out, especially in this endemic region . Left-siClassic symptoms of acute diverticulitis are fever, left lower abdominal pain and tenderness, in addition to an increase in markers of acute inflammation . In geneOn imaging, fistula can be detected either by direct (insertion of contrast through the fistulous tract) or indirect (the communication of the organs or by air detection in the fistulous tract) visualization. In colovesical fistula, only indirect contrast visualization is applicable. CT scan can portray endoluminal air, mural thickening, mucosal hyperemia, tethering of adjacent thick-walled bowel as well as the presence of pericolic fat streakiness . MagnetiColovesical fistulas caused by complicated acute diverticulitis are mainly treated by surgery. At first, medical therapy including bowel rest and empirical antibiotic therapy may be implicated in fistula of non-malignant origin. This indicates that it is extremely crucial to exclude malignancy in such cases. At present, laparoscopic surgery for colovesical fistula offers equal safety and feasibility as compared to open surgery, especially in a high volume laparoscopic colorectal surgery centre . Laparos4Pneumaturia and faecaluria are the common but distinct presences of a variety of diseases ranging from simple urinary tract infections to complicated diverticular diseases. This case report stressed the importance of thorough clinical history and physical examination aided by radiographic investigations to get an early diagnosis of diverticulitis. General practitioners and physicians should be aware of pneumaturia and faecaluria in clinical practice to reduce acute diverticulitis-related morbidity.Ethical approval was obtained from the local institution.NoneAAW - manuscript preparation, KSY - data collection. NKM - literature search, NA - literature search, ADZ - final review, FH - final reviewNo ethical clearance required as it only involves case report.Firdaus HayatiWritten informed consent was obtained from the patient for publication of this case report and accompanying images. Permission was also obtained from local administrators. A copy of the written consent is available on request.Not commissioned, externally peer-reviewedThe authors declare that there are no conflicts of interest.


A ruptured omental pseudoaneurysm is a rare cause of intra-abdominal hemorrhage. Herein, we present a case of bleeding ruptured omental pseudoaneurysm in a patient on systemic anticoagulation and successful treatment with surgery. A 72-year-old female on warfarin for atrial fibrillation presented with worsening abdominal pain. Cross-sectional imaging was obtained and was consistent with a large omental pseudoaneurysm (measuring 2.2 cm) as well as blood products within the abdomen. The patient was taken to the operating room where a pseudoaneurysm with evidence of active bleeding was identified. A diagnostic laparoscopy converted to exploratory laparotomy with partial omentectomy was performed. An omental pseudoaneurysm is a rare but potentially life-threatening cause of intra-abdominal hemorrhage. Given the risk of re-bleed, these lesions should be addressed promptly. In a facility that has the expertise, a catheter based approach with embolization may be considered, however, the mainstay of therapy should remain surgical resection. Multiple etiologies of intra-abdominal hemorrhage exist and include traumatic injury, rupture of visceral aneurysms, neoplasia or complications of anticoagulant therapy. Omental pseudoaneurysms are one of the rarer etiologies and can lead to rupture with a potential for life-threatening intra-abdominal hemorrhage . As omenA 72-year-old female with a past medical history of chronic obstructive pulmonary disease, gastroesophageal reflux disease, hyperlipidemia, coronary artery disease and paroxysmal atrial fibrillation on warfarin  presented to a referring facility emergency department with worsening abdominal pain throughout the day. The patient had taken warfarin for several years without previous complications. Furthermore, she had no history of trauma or spontaneous bleeding. Her initial vital signs were unremarkable. Significant laboratory values included a hemoglobin of 13.0 and an international normalized ratio (INR) of 3.57, whereas all other labs were within normal limits. Cross-sectional imaging was obtained and the computed tomography (CT) scan revealed a left omental lesion measuring 2.2 cm with arterial and portal/venous enhancement concerning for a complex pseudoaneurysm . ImagingUpon arrival to our facility, the patient remained hemodynamically stable. Her warfarin was reversed with vitamin K as well as factor eight inhibitor bypassing activity (FEIBA) resulting in a normal INR level. The surgical service was consulted and due to need for long term anticoagulation, moderate volume hemoperitoneum and concern for rupture, the patient was taken to the operating room (OR) for resection of the presumed ruptured omental pseudoaneurysm.In the OR, a laparoscopic approach was attempted given the patients stability. Upon insufflation of the abdomen, a moderate amount of hemoperitoneum was encountered. Further laparoscopic ports were placed to facilitate exploration. Although the site of the pseudoaneurysm could be seen, it could not be safely mobilized secondary to adhesive disease from previous abdominal wall hernia repairs. Furthermore, there was noted to be continued bleeding from the pseudoaneurysm. Conversion to an upper midline laparotomy facilitated direct visualization and finger pressure control of the bleeding pseudoaneurysm. The pseudoaneurysm within the omentum was mobilized and resected with an energy device and sent for pathologic evaluation. The abdomen was then irrigated and closed.Post operatively, the patient recovered uneventfully and was discharged home on post-operative Day 3, after re-initiation of her oral anticoagulation therapy. Final pathology revealed a vessel with blood clot, histocytes and giant cells in the wall, which was consistent with a ruptured pseudoaneurysm. At her 2-week follow up in the clinic, she was continuing to recover well.An omental pseudoaneurysm is a rare cause of intra-abdominal pain and hemorrhage and infrequently identified prior to definitive intervention . They arThere are no specific recommendations for the treatment of pseudoaneurysms of the greater omental artery. However, recent recommendations from the Society of Vascular Surgery are to treat gastric, gastroepiploic, gastroduodenal and pancreaticoduodenal artery aneurysms or pseudoaneurysms of any size . FurtherWhen diagnosed on initial imaging, there are several therapeutic modalities that can be employed. Within the last several years, there have been multiple case reports within the Japanese literature describing transcatheter arterial embolization as treatment for omental pseudoaneurysms , 3. The In about half of cases, patients may have their omental pseudoaneurysm initially misdiagnosed as an acute abdomen for another pathology, and only upon operative exploration are they correctly identified. In the other half of cases that are identified prior to intervention, less invasive techniques besides laparotomy can be considered , 3. As iNo specific recommendations exist for follow up or surveillance for resected greater omental pseudoaneurysms. For most visceral aneurysms, recommendations include one-time cross-sectional imaging to identify the presence of concomitant aneurysms. For embolized gastroepiploic lesions, recommendations for surveillance include imaging every 1–2 years, however, no recommendations exist following resection of these lesions . SimilarAn omental pseudoaneurysm is a rare but potentially life-threatening cause of intra-abdominal hemorrhage. Given the risk of re-bleed and rupture, these lesions should be addressed promptly. In a facility that has the expertise, a catheter based approach with embolization may be considered, however, the mainstay of therapy should remain surgical resection, especially in the setting of any sort of hemodynamic instability, concern for ongoing hemorrhage, or risk of underlying malignancy.


A tissue biopsy is crucial for a definitive diagnosis. Lymphocytic cholangitis is one type of cholangitis and is identified through histological findings. Treatment requires a long-term immunosuppressive drug. This article is the first report of multiple cyst-like hepatic lesions with histological findings of lymphocyte infiltration in a cat’s liver.CholangitisPlatynosomum fastosum), intestinal protozoa, and other helminth eggs. Praziquantel was prescribed for two distinct courses one month apart without obvious improvement of the hepatic cysts. An abdominal laparotomy and histopathological examination finally enabled diagnosis of cyst-like lymphocytic cholangiohepatitis of the liver tissue. Twelve weeks of oral prednisolone resulted in marked ultrasonographic improvement of the hepatic cysts. The liver parenchyma was heterogeneous and filled with multiple small anechoic cavities. Twenty-three months after ceasing the prednisolone, there was no recurrence of hepatic cysts.A 5-year-old female neutered domestic short-haired cat presented with abdominal enlargement. An abdominal ultrasound revealed that large multiple hepatic cysts with irregular walls, hypoechoic fluid, and internal septations occupied most of the liver parenchyma. Serum liver enzymes, bilirubin, and bile acids concentrations were within normal limits. A fecal examination using simple floatation and formalin-ether sedimentation techniques was negative for liver fluke ( Feline cholangitis/cholangiohepatitis is an inflammatory condition of two conjunctive organs: the gall bladder and the liver ,2. The pAlthough lymphocytic cholangitis/cholangiohepatitis is commonly found, cystic lesions of the hepatic parenchyma have not been described in cats with lymphocytic cholangitis/cholangiohepatitis. The present article represents the first report of clinical signs and characteristics of cyst-like lesions on the hepatic parenchyma with lymphocytic cholangitis/cholangiohepatitis in a cat.TM Hematology Analyzer, Sysmex, Lincolnshire, IL, USA) and an automatic chemistry analyzer . A complete blood count indicated regenerative anemia with a hematocrit of 16.4% : 30–45%), an increased mean corpuscular volume of 60.07 fL (RI: 39–55 fL), absolute reticulocytosis of 225,000/µL , and neutrophilia of 14,547 cells/µL   was brought to the Kasetsart University Veterinary Teaching Hospital with a 1-month history of progressive abdominal enlargement. Informed consent was obtained from the cat’s owner. The cat had indoor and outdoor access and was fed a commercial, complete, and balanced cat diet. The cat was alert and had hyporexia. A complete physical examination was performed and vital parameters were within normal limits. The clinical examination revealed that the cat had pale mucous membranes and moderate abdominal distension. Hematology and blood chemistry testing was performed using an automated hematology analyzer (Sysmex XN-1000ells/µL) . A bloodells/µL) . A high ®, Lyon Cedex, France), was negative. Doxycycline  was prescribed orally for suspected M. haemofelis because the cat had regenerative anemia and lived in an area where the tick-borne parasite is endemic, although the blood smear results were negative for hemoparasites.The results of a combination enzyme-linked immunosorbent assay (ELISA) test for the feline leukemia (FeLV) antigen and the feline immunodeficiency virus (FIV) antibody, using a commercially available FeLV–FIV rapid immunochromatography test assay (WitnessPlatynosomum fastosum), intestinal protozoa, and other helminth eggs. However, praziquantel  was prescribed for 4 days.The abdominal ultrasound was performed by the Thai Board of Veterinary Surgeons, sub-specialty of veterinary diagnostic imaging, using a real-time scanner  with a 13 MHz broadband linear transducer. An abdominal ultrasound identified multiple large hepatic cysts occupying most of the liver . The cysAfter 4 weeks of doxycycline administration, the cat’s weight was 5.5 kg. The cat was alert and had a normal appetite. Moderate abdominal distension was still found during the physical examination. Blood tests showed signs of improvement of the anemia, with a hematocrit of 27.5% (RI: 30–45%) and a decreased mean corpuscular volume of 35.99 fL (RI: 39–55 fL).Platynosomum fastosum infection were unlikely. Therefore, an exploratory laparotomy was performed. Anesthesia was induced with a slow IV infusion of propofol  and then maintained with isoflurane . Intramuscularis injection of morphine sulphate  was used for analgesia. Multiple large hepatic cystic lesions occupied the liver parenchyma and could not be removed. There was a small area of non-cystic liver parenchyma. Therefore, incisional biopsies of the liver and hepatic cyst walls were collected for histopathology. The cat’s anesthetic recovery went well. Intravenous fluid therapy using 0.9% NaCl  and antibiotic therapy using amoxicillin-clavulanate intravenously  was continued for postoperative care. Tramadol  was administered for analgesia. The cat was bright with a good appetite and was discharged 72 h postoperatively, with the sutures removed 7 days later.One month after the first dose of praziquantel administration, the cat received the second dose of praziquantel in a similar fashion to the initial dose. However, there was no noticeable improvement of the abdominal enlargement and hepatic cysts as determined by a clinical examination and an ultrasonographic evaluation. Parasitic cysts due to ®, Berlin Pharmaceutical Industry Co., Ltd., Bangkok, Thailand) and silymarin , were also administered.Three Thai board-certified pathologists analyzed the samples. Histopathology of the liver demonstrated lymphocytic cholangiohepatitis A,B. MixeAfter 12 weeks of prednisolone administration, the abdominal distension had disappeared. In addition, improvement of the hepatic cysts was detected by abdominal ultrasonography. The abdominal ultrasound demonstrated a heterogeneous liver parenchyma filled with irregular, small anechoic cavities on the left lobe of the liver A,B. The Twenty-three months after stopping prednisolone, the cat was doing well, was active, and had a good appetite. An ultrasonographic examination showed no recurrence or progression of the hepatic cysts C,D. The The hepatic cyst-like lesions in this present case were diagnosed as lymphocytic cholangiohepatitis. In general, biochemical parameter abnormalities of cats with lymphocytic cholangitis/cholangiohepatitis are often revealed by an increase in one or more liver enzyme activities ,3. InterTrue hepatic cysts are fluid-filled cavities lined with epithelium; however, the pathogenesis is not well defined . The cysClinical manifestation of liver fluke infestation can be hepatic cysts. Detection of liver fluke eggs from fecal examination may be missed because of the intermittent egg shedding and low number of eggs in feces . CytologOn occasions, hepatic cysts become more complex lesions, characterized by irregular walls, septations, internal debris, or varying degrees of echogenic content. In these cases, neoplasia, hematoma, abscess, necrosis, inflammation, parasites, or toxins must be included in the differential diagnosis ,31. In oLymphocytic cholangitis/cholangiohepatitis in cats is characterized as the infiltration of small lymphocytes in the portal areas, with varying degrees of fibrosis and biliary proliferation. The pathogenesis of lymphocytic cholangitis/cholangiohepatitis was proposed to be involved with an autoimmune mechanism and bacterial infection ,32. UltrTo the authors’ knowledge, complex cyst-like lesions of the liver parenchyma of a cat with lymphocytic cholangitis/cholangiohepatitis have not been reported. The cyst-like lesions contained the areas of necrotizing hepatitis, tissue hemorrhage, and eosinophilic fibrinous proteinaceous materials. The cause of hepatic cyst-like lesions in this case remains unclear. The hepatic cyst might be the primary cause. The gradual distention of the cysts might induce biliary obstruction, which promotes bacterial infection followed by cholangitis ,34,35,36Treatment with anti-inflammatory dosages of prednisolone clearly improved the hepatic cyst-like lesions in this study, possibly because the cat was diagnosed early and showed normal liver function. We suggest histological analysis should be performed in cats with complex hepatic cysts that involve large parts of the hepatic parenchyma.We have demonstrated the first case of feline lymphocytic cholangiohepatitis that presented with cyst-like structures in the liver. Histopathological analysis is important for diagnosis and leads to successful treatment with prednisolone.


A healthy 42‐year‐old man presented to the hospital because of chest radiography performed during a medical checkup revealed a tumor. Contrast‐enhanced computed tomography showed a tumor of 5 cm diameter just above the right diaphragm with blood flow from the portal vein. The patient was diagnosed with accessory liver lobe (ALL). No finding suggested malignancy, and he is being followed up. ALLs are usually found in the abdominal cavity, but they can also be found in the thoracic cavity. Although ALL is rare, it should be considered in patients presenting with intrathoracic tumors. A healthy 42‐year‐old man presented to the hospital because chest X‐ray taken during a medical checkup had revealed a tumor. He was diagnosed with accessory liver lobe (ALL). No findings suggested malignancy, and he is being followed up.Although ALL is rare, it should be kept in mind in patients presenting with intrathoracic tumor. ALL was first described in 1767.2A healthy 42‐year‐old man presented to the hospital because his chest radiograph obtained during a medical checkup revealed a mass overlapping the right diaphragm Figure . He was 3The first lesson of this case is the importance of proper detection of tumors in contact with the diaphragm on a chest radiograph. This is because superimposed normal structures covered by the diaphragm are the major contributing factor to overlooked lesions.The authors have stated explicitly that there are no conflicts of interest in connection with this article.All authors meet the ICMJE authorship criteria.The patient has provided free written informed consent for the publication of this manuscript.The patient provided written informed consent.


Abdominoscrotal hydrocoeles (ASH) are an increasingly rare form of hydrocoeles. They can present in any age group; however, they are more commonly reported in the paediatric population. Although not yet confirmed, the aetiology of ASH has been under scrutiny for the past two centuries, and scarcity of reported cases hinders this process. Clinical examination is oftentimes sufficient to make the diagnosis; however, confirmatory ultrasonography is recommended. Although old reports favoured a more conservative approach with watchful waiting, the risk of serious secondary complications is high, and surgical intervention is hence considered standard of care. Different approaches have been described, each with their own benefits, with minimally invasive surgery becoming more prevalent as of late. A case of a 10-month-old boy with bilateral ASH treated with a laparoscopic technique is presented. A hydrocoele is a cystic mass filled with fluid that develops between the two layers of the tunica vaginalis, the serous membrane that covers the testes . HydrocoNotably, abdominoscrotal hydrocoeles (ASH) are renowned for being the rarest of all hydrocoele forms, and they typically involve a dumbbell-shaped collection which extends from the scrotum into the abdomen via the inguinal canal . ContrarWe herein present a case of a 10-month-old boy with a bilateral abdominoscrotal hydrocoele who underwent laparoscopic surgical repair.A 10-month-old boy presented to the paediatric surgery outpatient clinic as a referral from his paediatrician. Prior to this presentation, the patient had been seen by his paediatrician and was found to have bilateral testicular swelling since birth. Upon being examined by the paediatrician, the swelling was found to be approximately 5x4cm in size and was translucent in nature. The infant was thus diagnosed with a bilateral hydrocoele, and expectant management was advised, with watchful waiting until one year of age. He was otherwise fit and well, with no significant past medical or surgical history.Over the course of the next few months since paediatric assessment, the infant’s testicular swelling dramatically increased in size, warranting a review. Despite the patient being haemodynamically stable with no alarming local, gastrointestinal or urinary symptoms, the drastic increase in size of the swelling prompted a referral to paediatric surgeons for further assessment and investigation.On examination in the paediatric surgery clinic, the 10-month-old boy was found to have remarkable swelling of the scrotum, with the left side being positive for translucency and the right side being negative for the same test. Neither of the testicles was palpable at this point in time. General examination was otherwise unremarkable. Marked lymphoedema of the phallic skin and foreskin was also noted, as can be seen in Figure The aforementioned findings warranted further investigation with scrotal ultrasonography, which revealed massive bilateral ASH  with a characteristic dumbbell appearance, homogenous anechoic content, and positive Doppler blood flow detected in both testes with no evidence of bowel herniation in either of the inguinal canals. The patient was thus scheduled for a laparoscopic procedure and admitted to the day-case unit. Laparoscopic procedure was performed under general anaesthesia with right-sided ligation of patent processus vaginalis (PPV) and hydrocoele percutaneous aspiration. Left-sided abdomino-inguinal hydrocoele was repaired in a similar fashion with aspiration and deroofing, as well as PPV ligation. Right-sided hydrocoele aspiration was quantified at 15 millilitres. Aspiration of the left-sided hydrocoele amounted to 100 millilitres. Bilateral PPV closure was performed using the LAPIRS  technique. Figure Whilst in the operating theatre, the patient also underwent circumcision upon patient parental request (as is customary in Jordan). It is worth noting that circumcision procedure was done irrespective of diagnosis and was not part of active management of the patient’s presenting condition. The procedure was performed successfully with no intraoperative complications, and appropriate haemostasis was ensured throughout. Post-operative recovery was satisfactory, with the patient being admitted overnight for prophylactic antibiotics and fluids. The patient was deemed fit for discharge the following day and has since improved remarkably with no signs of recurrence on routine follow-up.A hydrocoele, in all its many shapes and forms, although frequently benign, can in many instances be the manifestation of a more lethal underlying condition. ASH in particular, being the least common variant, is the type known to most often present with significant complications if not rectified in due course .A systematic review performed in 2018 by Gadelkareem included 320 studies, of which 299 were single case reports and 21 were case series. The total number of ASH cases was thus reported to be 579 cases, with 360 cases reported in paediatric patients and the remainder reported in adults. In paediatric cases, rates of ASH were found to range from 0.18% to 3.1% of all types of hydrocoeles. This systematic review described all cases between 1777 and2017, highlighting the rarity of the condition .Although first identified in 1777 by Parcival-Pott, it was not until 1834 that Dupuytren postulated an aetiological explanation for ASH ,3. HowevIdentifying ASH as a diagnosis can, in many cases, be a challenge. Generally speaking, it should be suspected when a scrotal swelling is found in conjunction with an abdominal swelling that is localised around the area of the deep inguinal ring . In the Ultrasonography is an easily accessible and commonly used method for further evaluation and is generally recommended for verification of the diagnosis . DynamicImportant differential diagnoses to rule out include spermatic cord lymphangioma, testicular tumour, severe cases of hydronephrosis that extend into the inguinal area, bladder diverticulae and pelvic neuroblastoma .In comparison to other types of hydrocoeles, once a diagnosis of ASH is confirmed, intervention must be scheduled. This is due to the increased chances of secondary complications if left untreated. Complications of ASH include impacted spermatogenesis, sexual dysfunction and infertility, rupture and, consequently, haematocoele formation, hydronephrosis, appendicitis, lymphoedema, cryptorchidism, and malignant transformation to name a few ,2,10.Although different perspectives on the most suitable means of managing ASH exist, surgical intervention remains the standard of care for this condition . Whilst Overall, the surgical procedure is considered to be a difficult one due to the fact that in many cases the hydrocoele attaches itself to the cord structures along with a thickened tunica vaginalis, which can be difficult to access regardless of technique. Aspiration of the fluid prior to dissection of the membrane can thus help facilitate easier access . The useASH is a rare form of hydrocoeles. Knowing the embryological origin as well as the common presenting features is pertinent to making an accurate diagnosis. Despite prevalent controversy over the true aetiology of this condition, simple manoeuvres employed during physical examination, alongside a confirmatory ultrasonography, can in most cases be sufficient to make a diagnosis. In doing so, appropriate surgical management can ensue and serious complications are thus avoided.


Sebaceous adenoma is an extremely rare tumor located in the parotid gland. In the English literature, less than 10 cases have been reported. Sebaceous adenoma represents 0.5% of all monomorphic adenomas. The authors are presenting a case of sebaceous adenoma of the parotid gland in a 65-year-old female who presented a mass on the left parotid area that had been gradually enlarging for one year without symptoms of pain. On imaging (ultrasound), a well-defined mass lesion in the left parotid area was seen. Histopathological findings were consistent with sebaceous adenoma. Surgical excision is curative. The prognosis is excellent, with a low recurrence rate. The present case report will increase the awareness and possibility of this rare tumor occurring at an unusual site, thereby avoiding any chance of misdiagnosis. Sebaceous adenoma is a rare benign neoplasm derived from epithelial tissue that displays sebaceous differentiation ,2 and waHerein, we report a case of a 65-year-old woman with sebaceous adenoma of the parotid gland; it is extremely rare insofar as being limited to a few cases reported in the medical literature. After that, we discuss its diagnostic features, including clinical, imaging, and histopathologic findings, with the aim of increasing awareness of this infrequent entity.A 65-year-old woman presented with a slowly growing palpable mass of the left parotid gland, which was first seen one year earlier. There was no history of pain, bleeding, trismus or discharge from swelling during the course of enlargement, history of fever, or altered salivary flow. She had a 40 years history of smoking. Associated diseases included arterial hypertension and New York Heart Association (NYHA) class III. Physical examination of the left parotid gland disclosed a soft, non-mobile parotid gland tumor of 4 cm in diameter and has no tenderness to palpation. The tumor was covered by a normal-appearing skin not attached to the deeper tissues. The overlying skin appeared with no evidence of scar or discoloration. The skin over the tumor was mobile and pinchable. There was no evidence of cervical lymphadenopathy or neurological affection.An ultrasound scan (US) revealed a parotid tumor with well-defined margins, isoechoic texture, and no adenolymphomas . Chest XBased on the history, clinical examination, and ultrasonography findings, a provisional diagnosis of Warthin’s tumor was made. A left superficial parotidectomy with facial nerve preservation was performed. The patient was discharged 7 days after her surgical procedure without any complications.Histopathology reported a well-circumscribed tumor of 3.2 × 2 × 1.6 cm, surrounded by normal parotid tissue, white, yellowish color, and some cystic spaces. The cut surface was homogeneous, with small cystic spaces a,b.Histologically, paraffin sections stained with hematoxylin and eosin showed a microcystic appearance with numerous sebaceous glands, with various dimensions, embedded in stromal fibrosis a,b. OccaThe patient’s postoperative course was uneventful; until now (4 months later), she has remained without evidence of recurrence. The patient is still under follow-up.Although most commonly associated with hair follicles, ectopic sebaceous glands can be identified independently. These lesions can be found mainly on the eyelids and oral mucosa . OccasioDespite the common occurrence of sebaceous differentiation in salivary glands , sebaceo® and Web Of Science®, published during the all-time topic, screening for the keyword “sebaceous adenoma of the parotid gland”.We performed a search on two databases, PubMedAfter the screening, we found a few reports concerning sebaceous adenoma located in the parotid gland .Following the analysis of the all-time literature, the current case is the tenth reported case of sebaceous adenoma with localization in the parotid gland. The case presented showed differentiated sebaceous lobules accompanied by a fibrous stroma.Thus, de Vicente-Rodríguez reported the histopathological appearance in a case of sebaceous adenoma located in the parotid gland . Foote aSebaceous adenomas of the skin sites may be associated with nonpolyposis colorectal cancer, a condition known as Muir–Torre Syndrome ,24, an aGenerally, sebaceous adenomas in the parotid gland are painless and slow-growing well, encapsulated lesions that do not infiltrate the surrounding normal tissues, with no facial nerve involvement. The tumor size typically ranges from 0.5 to 5 cm in diameter. They histologically appear as white-yellowish nodules that have a microcystic pattern, with abundant well, differentiated sebaceous glands of variable size ,27. The Besides clinical examination, ultrasonography (US), computed tomography (CT), and magnetic resonance imaging (MRI) are the most common radiological procedures for the diagnosis of tumor-like lesions of the salivary glands. According to Haynes et al., the initial diagnostic test for salivary gland tumors should be computed tomography (CT), which offers information regarding the mass’s size, extent, location, content, and consistency . HoweverThere is no consensus about using fine-needle aspiration biopsy (FNAB) in major salivary gland tumors . AccordiDifferential diagnosis should include other benign parotid tumors with solid mass with cystic change patterns .Usually, these benign tumors can be removed, and they are not likely to recur if adequately excised. Since there is no evidence for efficacy, radiation therapy should not be considered an alternative to surgery in sebaceous adenoma. Thus, curative surgical excision is the treatment of choice.The authors described an extremely rare case of sebaceous adenomas located in the parotid gland, which might help to enrich the clinical spectrum of this rare site. This case illustrates that sebaceous adenoma should be considered in the differential diagnosis of every soft tissue tumor in any major salivary gland, especially when it occurs in the parotid gland region.


A six-year-old boy visits a general practitioner due to diarrhea and abdominal pain with a moderate fever of up to 39 °C for 2 days. Treatment is initiated; however, the recurrence of abdominal pain is observed. Physical examination of the child at the emergency department reveals abdominal guarding and visible, palpable, painful intestinal loops in the left iliac and hypogastric regions—this is referred to as an ‘acute abdomen’. An X-ray shows single levels of air and fluid indicative of bowel obstruction. Ultrasound reveals distended, fluid-filled intestinal loops with diminished motility. The intestinal wall is swollen. Laboratory tests indicate increased inflammatory indices. Contrast-enhanced computed tomography examination of the abdominal cavity and lesser pelvis shows intestinal dilation. The loops were filled with liquid content and numerous collections of gas. The patient is qualified for a laparotomy. An intraoperative diagnosis of perforated gangrenous appendicitis with autoamputation was made. In addition, numerous interloop and pelvic abscesses, excessive adhesions, signs of small intestine micro-perforation, and diffuse peritonitis are found. The patient’s condition and laboratory parameters significantly improve during the following days of hospitalization. Despite the implementation of multidirectional, specialized diagnostics in the case of acute abdomen, in everyday practice we still encounter situations where the final diagnosis is made intraoperatively only. An acute abdomen is a medical condition that occurs unexpectedly, progresses rapidly and is commonly life-threatening. The underlying causes include diseases of the liver, stomach, intestines, and genitourinary system. Intestinal obstruction accounts for approximately 15% of all emergency visits for acute abdominal pain and is typically manifested by the following three symptoms: abdominal pain, vomiting, gas, and stool retention . Other cA six-year-old boy visited a general practitioner (GP) due to diarrhea and abdominal pain with a moderate fever of up to 39 °C for 2 days. Based on a medical interview and physical examination, azithromycin was prescribed orally at a standard dose. The patient’s condition clearly improved. No imaging examinations were performed at that time. Four days following the end of treatment, the recurrence of abdominal pain, which was increased by changing the body’s position, was observed. The child was admitted to the emergency department where he was diagnosed with ‘acute abdomen’. The SARS-CoV19 antigen test was negative. Physical examination revealed abdominal guarding and visible, palpable, painful intestinal loops in the left iliac and hypogastric regions. A successful stool enema was conducted. Subsequently, diagnostic imaging was performed, including abdominal X-ray using Carestream Evolution Plus  and ultrasonography (US) using Philips Epiq 5 . Single air-fluid levels indicative of bowel obstruction were detected on X-ray a. The USThe patient was referred to the Department of Pediatric Surgery and Traumatology. Due to developing sepsis and symptoms of multiple organ dysfunction syndrome (MODS) in the patient, an extensive panel of laboratory tests was performed, which indicated increased inflammatory indices such as PCT, CRP, ESR, ferritin, the level of leukocytes and neutrophiles . It is wBased on repeated physical examination, which revealed diffuse peritoneal signs, and all the previous laboratory and diagnostic imaging findings, the patient was qualified for surgical treatment, i.e., laparotomy. An intraoperative diagnosis of perforated gangrenous appendicitis with autoamputation was made. In addition, numerous interloops and pelvic abscesses, excessive adhesions, signs of small intestine micro-perforation, and diffuse peritonitis were found.Although the stool culture was negative for Yersinia, the blood methicillin-resistant coagulase-negative Staphylococcus (MRCNS) epidermidis, and non-ESBL (extended-spectrum beta-lactamase)-producing Escherichia coli were detected in the blood and pus cultures, respectively.A follow-up US examination indicated mild edema of the bowel wall and peri-intestinal fat. Moderately dilated (lumen width approx. 15–20 mm), fluid-filled intestinal loops  with diminished motility, were observed as well . The patDiagnosis of appendicitis in children appears to be a well-known topic but may still be an issue due to non-specific clinical symptoms and variable, inconclusive imaging studies. Dilatation of bowel plexuses and obstruction require careful evaluation, and it is difficult to determine the cause. The incidence rate of chronic appendicitis is about 1.5% in patients who present with symptomatology similar to acute appendicitis . AnotherIn summary, appendicitis is a condition that can be life-threatening if left untreated. The underlying inflammation may result in numerous complications and a significant deterioration of the patient’s health. Chronic appendicitis, especially in children, often has a non-specific image; therefore, detailed laboratory and imaging diagnostics are particularly important. In the above-described case of a six-year-old boy, clinical examination of the patient, ultrasound, and CT of the abdominal cavity suggested intestinal obstruction. However, based on the clinical and imaging diagnostics performed, its cause could not be determined. Only intraoperatively gangrenous perforated appendicitis with self-amputation, as well as multiple abscesses were diagnosed. Despite the implementation of multidirectional and specialized diagnostics in the case of acute abdomen, in everyday practice we still encounter situations where the final diagnosis is made intraoperatively only.


Granular cell tumours (GCTs) of the pancreas are mostly benign and exceptionally rare, with no unique identifying radiological features. Following a case discussion of a patient with GCT, a comprehensive review of available literature was conducted to identify the common diagnostic features associated with GCT.Following a case report identified in our institution, a systematic review was conducted by two authors in accordance with Preferred Reporting Items for Systematic review and Meta-Analysis protocols (PRISMA) guidelines. Databases MEDLINE, EMBASE, Scopus, World of Science, and grey literature were searched on August 2021. Inclusion criteria were histopathology diagnosed granular cell tumour of the pancreas.A 37-year-old male presented with 1 month of abdominal pain and an MRI demonstrating a dilated main pancreatic duct, distal parenchymal atrophy, but no focal lesion. Repeat MRI at 6 months re-demonstrated similar findings and subsequent endoscopic ultrasound was suspicious for main duct IPMN. Following multidisciplinary team discussion, a spleen-preserving distal pancreatectomy was performed. Histopathology demonstrated granular cell tumour with cells diffusely positive for S100 and no malignant transformation.11 case reports were identified in the literature with diagnosis confirmed on tissue histopathology based on positive immunohistochemical staining for S-100 protein. Eight patients presented with gastrointestinal symptoms with abdominal pain the main presenting complaint (50%). 10 patients underwent CT with portal venous contrast and all underwent endoscopic examination. Imaging findings were similar in five studies for EUS which demonstrated a hypoechoic lesion with homogenous appearance. On non-contrast CT GCT was iso-enhancing, and with portal venous contrast demonstrated hypo-enhancement that gradually enhanced on late phases. Pre-operative diagnosis of pancreatic carcinoma was described in six cases based on imaging and biopsy, resulting in progression to surgical resection. Nine patients were managed surgically and no complications identified on follow-up (6–52 months).The currently proposed management pathway includes EUS with biopsy and CT, and surgical resection recommended due to malignancy risk. Improved sample collection with EUS-FNA and microscopic assessment utilising S-100 immunohistochemistry may improve pre-operative diagnosis. Limitations include rare numbers in reported literature and short follow-up not allowing an assessment of GCT’s natural history and malignancy risk. Additional cases would expand the current dataset of GCTs of the pancreas, so that surgical resection may be avoided in the future. Granular cell tumours (GCT) are rare tumours of Schwann cell origin and, whilst they are most commonly benign, malignancy has been reported in less than 2% of cases . First rA 37-year-old male presented with a 1-month history of intermittent, mild upper abdominal pain radiating to the back. An outpatient abdominal ultrasound demonstrated a pancreatic lesion suspected to be an intra-papillary mucinous neoplasm (IPMN), an uncommon diagnosis in this age group. Initial magnetic resonance imaging (MRI) showed no evidence of an enhancing mass though the main pancreatic duct (MPD) was dilated up to 6 mm. There was mild enhancement of the distal pancreatic body and tail in the post-gadolinium images, suggestive of atrophic changes secondary to main duct IPMN. The patient was observed for 6 months and a repeat MRI showed similar findings with unchanged dilatation of the MPD and subjacent pancreatic parenchymal atrophic changes Fig.  and b, bA systematic review was conducted in accordance with Preferred Reporting Items for Systematic review and Meta-Analysis protocols (PRISMA) statement. Review was registered in PROSPERO ID CRD42022356253. Databases MEDLINE, EMBASE, Scopus, World of Science, PubMed, and the grey literature were searched using MeSH terms with Boolean operators “granular cell tumour” AND “pancreas” up to August 2021. Inclusion criteria included human patients with primary granular cell tumour of the pancreas confirmed on histology. Exclusion criteria were patients without histological diagnosis of GCT defined by S100 positive staining on immunohistochemistry. Full criteria in Table Reference lists and grey literature were examined to broaden search strategy and capture studies. No language, regional, or chronological restrictions were made. Study quality and risk of bias was assessed with the tool by Murad  to evaluTwo authors (SM and KT) assessed studies for inclusion and their quality, with disagreements resolved using an independent third author (KK) by consensus.Two independent authors (SM and KT) extracted data from studies into Microsoft Excel with outcomes collected including patient background, clinical presentation, investigations, radiological findings, histopathology findings, management, morbidity, mortality, and follow up. Descriptive statistics were used for reported outcomes, with dichotomous variables recorded as percentages and continuous variables as median. Qualitative outcomes for radiology findings were interpreted through narrative synthesis. Statistical analysis of Point-Biserial correlation was performed with IBM SPSS Statistics version 28 .The search strategy is detailed in the PRISMA flowchart Fig. . 614 stuQuality of papers assessed demonstrated moderate quality in 5 papers and high in 6 papers Table . Due to 11 case reports were identified with characteristics in Table Seven patients presented with gastrointestinal symptoms, with abdominal pain the main presenting complaint in 45.5% of cases. Two patients were asymptomatic and discovered as incidental findings radiologically , 19 althExcept for the 1975 study , all patImaging findings Table  were simp = 0.985). GCT size and abdominal symptoms showed a correlation 0.718 but similarly non-significant (p = 0.718).MPD dilatation was present in 7 patients (64%), of which all 7 patients with gastrointestinal symptoms had MPD dilatation. Point-biserial correlation of GCT size and MPD dilatation showed a negative correlation − 0.07, however, was non-significant (10 patients were managed surgically and 1 non-operatively. Two patients had distant disease , 20 of wThere have only been eleven reported cases of GCT of the pancreas, and with this case report, twelve cases with findings and comparisons included in this systematic review Table .GCTs are more commonly found in adults, and more than 50% of them are located within body of the pancreas , 21. AltThe MRI findings of GCT have been previously reported, although without much consistency in its radiological features. In our study, MRI was unable to identify a well-defined mass; however, there was distal main duct dilatation similar to other case reports , 13, 20.Despite the utility of FNA under EUS guidance in the work-up of pancreatic masses and diagnosis of PDAC, its utility in the diagnosis of pancreatic GCT is unclear. This is arguably due to the limited sample that is obtained from an FNA, preventing from an accurate diagnosis. There have been two cases that have been successfully diagnosed using EUS , 19, of Multiple granular cell tumours have been reported in association with syndromes associated with aberrant RAS/MAPK signalling including Noonan Syndrome and neurofibromatosis –31. HoweLimitations of this review owe to the rarity of this disease process within the published literature. Pancreatic GCT is an extremely rare pathology and the diagnostic obscurity of this condition still remains, despite the few case studies that have been reported to date. Subgroup analysis to examine mortality, malignancy, or the natural history could not be performed due to lack of data and short follow up duration. A meta-analysis was planned however not performed as deemed inappropriate due to limited data and clinical and statistical heterogeneity. Study heterogeneity exists owing to the published literature’s broad timespan of 44 years, which is reflected in the distinct change and usage of contemporary imaging modalities EUS and MRI. Quality of review’s case reports was moderate-high; however, the certainty of evidence assessment is low owing to scarce literature limited to case reports.Most studies did not elaborate on the decision-making process to proceed with operation, often the presumed reason being suspicion for malignancy. Thus, a comparison could not be performed to non-operative management only performed in one case, which discourages strong recommendations to pursue non-operative management and surveillance. Despite being a predominantly benign tumour with no cases of malignancy reported, a reported 32% risk of recurrence with malignant GCT exists . WithoutThus, we recommend initial radiological investigations with MRI and EUS, accompanied by FNA biopsy to confirm diagnosis. In lieu of confirmed diagnosis of GCT, we suggest multidisciplinary discussion prior to proceeding to radical resection due to diagnostic uncertainty encompassing other sinister lesions such as IPMN or malignant GCT.In conclusion, the majority of GCTs of the pancreas are only diagnosed on histopathological examination of the resected specimen. GCT of the pancreas should be considered as a diagnosis during the work-up of pancreatic masses and in the presence of pancreatic ductal dilatation. Further improvement in the diagnostic tools and techniques such as EUS may assist in making the correct diagnosis preoperatively and may alter the course of management for the patient. Although surgical resection is currently recommended for GCTs due to malignancy risk, the natural history of this condition in the pancreas is still unclear due to its infrequency. Additional cases would expand the currently available dataset of GCTs of the pancreas.