PDGFRB have been identified as the most common molecular defect in familial IM.Infantile myofibromatosis (IM) is the most common cause of multiple fibrous tumors in infancy. Multicentric disease can be associated with life-threatening visceral lesions. Germline gain-of-function mutations in PDGFRB-driven IM with multiple tumors at different sites, including intestinal polyposis with hematochezia, necessitating temporary chemotherapy.We here describe an infant with PDGFRB-driven IM is clinically challenging due to its fluctuating course and multiple organ involvement in the first years of life. Early molecular genetic analysis is necessary to consider tyrosine kinase inhibitor treatment in case of aggressive visceral lesions. Infantile myofibromatosis (IM) is a rare neoplastic condition with a reported incidence of 1 in 150,000 live births, but one of the most common causes of fibrous tumors in infancy . A detaiPDGFRB gene, encoding for the PDGFRb tyrosine kinase, which is mainly expressed in cells of mesenchymal origin, are a frequent cause of sporadic or familial IM [PDGFRB mutation in IM and has been shown to constitutively activate the PDGFRb receptor, eventually leading to cancer development [Activating somatic and germline point mutations in the ilial IM –10. Actiilial IM . The mutelopment .PDGFRB mutation c.1681C>T, with a remarkable clinical course in terms of spatial and temporal distribution of lesions, including intestinal manifestation with indication for chemotherapy. This case illustrates the challenges the clinician can be faced within the management of multicentric IM.We here describe a familial case of infantile myofibromatosis, harboring the characteristic Our patient is a female child that was born full-term after an uneventful pregnancy. Immediately after birth, a firm non-tender soft tissue mass beside the lumbar spine was noted. Sonographic assessment showed a well-circumscribed, vascularized soft tissue mass, measuring 3.6 × 2.8 × 1.7 cm, localized in the right paraspinal muscles. MRI at the age of 10 days demonstrated inhomogeneous T2 hyperintensity and contrast enhancement of the lesion . Cow’s milk protein allergy was initially suspected, but elimination diet of the nursing mother did not lead to cessation of rectal bleedings. Colonoscopy was performed at the age of 6 months, and at least fifteen polyps were detected in the transverse, descending, and sigmoid colon, with sessile morphology and a maximum size of 1.5 cm Fig. a. As no 2 once weekly over 3 weeks and actinomycin-D 1.5 mg/m2 and cyclophosphamide 20 mg/kg each as a single dose on day 1.Because of symptomatic polyposis with intermittent hematochezia and consecutive anemia, we decided to treat the child with a chemotherapeutic regimen of vincristine, actinomycin-D, and cyclophosphamide (VAC), according to the CWS guidance for non-resectable aggressive fibromatosis . Each VAAfter initiation of chemotherapy, no further episodes of hematochezia could be noted. Endoscopic reassessment after three VAC courses showed a decrease in size of polyps, so we decided to continue chemotherapy. After six VAC courses, we also noted a significantly decreasing number of polyps, as well as complete remission of the intramuscular lesions, so we then decided to omit further chemotherapy. Follow-up management was supplemented by endoscopic assessments every 3 months for early detection of progressive polyposis.Further disease course in our patient was characterized by a fluctuating picture. While intestinal polyposis was endoscopically undetectable 1 year after omission of chemotherapy, two new superficial lesions in the occipital region with a maximum size of 1 cm had emerged at the age of 15 months. These lesions were located to the tabula interna of the skull bone by means of MRI, clearly delineated from the intracranial space by the cortical bone. Two months later, while these two lesions already showed regression in size, another lesion in close proximity to the confluens sinuum, presumably also originating from the skull bone, was newly detected Fig. e. Due toPDGFRB gene (transcript ID NM_002609.3) was found in DNA of patient’s lymphocytes. Segregation analysis identified the mutation in the child’s father and her half-brother as well.Family history revealed that both the child’s father and one half-brother from the father’s side had spontaneously remitting subcutaneous nodules in infancy, with the half-brother being histologically diagnosed with “cutaneous leiomyoma.” In context of the positive family history and to explore the possibility of targeted therapy in case of relapse, germline genetic analysis was initiated. The characteristic missense mutation c.1681C>T in exon 12 of the This case of infantile myofibromatosis showed a remarkable disease course in terms of spatial and temporal distribution of lesions, necessitating chemotherapy due to symptomatic visceral involvement. Critical lesions in our patient were located in the gastrointestinal tract leading to hematochezia and the skull with proximity to the confluens sinuum.There are several reports of involvement of the gastrointestinal tract in IM in the literature. Its clinical manifestations most often include chronic diarrhea, poor feeding, and weight loss, but also can comprise acute life-threatening events such as bowel obstruction, perforation, or intussusception –18. OngoPDGFRB-driven IM arise temporally separated during the first years of life, with each lesion showing individual growth characteristics. Growth and regression of tumors can occur simultaneously in various tissues during this vulnerable period. However, tumor development and growth seems not to be strictly restricted to early childhood, as there are reports of adolescents and young adults with PDGFRB-driven IM, in whom tumor manifestations recurred or first occurred at a later age [PDGFRB-driven IM [PDGFRB germline mutation. However, molecular mechanisms triggering tissue- and age-specific tumor growth, regression, or recurrence in PDGFRB-driven IM largely remain obscure to date. At the time when diagnostic and treatment decisions had to be made in our patient, no specific guidelines existed to support decision-making, so management was mainly based on the individual clinical course. Recently, expert recommendations for the diagnosis and surveillance of patients with IM were published [PDGFRB germline mutations [CNS involvement is even much rarer in IM, but can lead to brain or spinal compression –21. Our ater age . Al Qawariven IM . These rublished . As all utations .PDGFRB mutations have been identified in most patients with multicentric IM and mainly seem to affect children. In one study, PDGFRB mutations were found in 9/15 children less than 2 years of age with multicentric IM, but not in adults with solitary myofibroma. It has therefore been proposed that PDGFRB-driven multicentric IM should be seen as a distinct entity of soft-tissue neoplasms in childhood [PDGFRB gene, leading to the substitution of arginine to cysteine at codon 561 (p.Arg561Cys). This mutation has been found recurrently in cases of familial IM. In fact, 17 out of 21 affected families, in whom a heterozygous PDGFRB germline variant was found, were reported to carry the p.Arg561Cys variant. Clinical information is available for 19 individuals with multicentric disease from these 17 families [PDGFRB gene was detected  be of diagnostic value in a histologically challenging disease and (2) offer the opportunity for molecular targeted therapy in patients with aggressive tumor expansion. Therefore, genetic counseling is nowadays of utmost importance in diagnosis, prognosis, and therapy guidance in IM. However, more data are needed to define genotype-phenotype correlations and thus help in clinical decision-making and prognostication for patients with IM tumor predisposition.In conclusion, we here describe a case of familial


Transethmoidal encephaloceles are rare and most commonly present at birth with congenital abnormalities, cerebrospinal fluid rhinorrhea, or visual symptoms. Here, we report the case of a 43-year-old presenting with longstanding headache, blurry vision, anosmia, and rhinorrhea. Magnetic resonance imaging confirmed a transethmoidal encephalocele. The patient underwent craniotomy for resection of the encephalocele and repair of the cribriform defect. The postoperative course was uneventful, and the patient was discharged home with the resolution of rhinorrhea and headache. This report highlights a rare case of primary transethmoidal encephalocele undiagnosed until adulthood despite longstanding symptoms and successful treatment with resolution of symptoms. Encephaloceles are herniations of brain tissue through a skull defect. Comprising only 1-3% of encephaloceles, transethmoidal encephaloceles are among the rarest . While tIn this case report, we describe a rare case of primary transethmoidal encephalocele, undiagnosed until adulthood despite longstanding symptoms and successful treatment with resolution of symptoms.A 43-year-old male with a body mass index (BMI) of 35 presented with a lifelong history of headache, blurry vision, anosmia, and rhinorrhea, but no reported history of significant trauma. The patient was previously worked up for bacterial sinusitis and allergic rhinitis and underwent unsuccessful trials of antibiotics, antihistamines, and intranasal steroids. The examination was remarkable for anosmia and visual impairment but no craniofacial abnormalities. Computed tomography and magnetic resonance imaging showed a 36 × 35 × 33 mm encephalocele herniating into the left ethmoids and nasal fossa with bony remodeling. There was complete opacification of the left frontal sinus and near-complete opacification of bilateral maxillary sinuses Figure .The patient underwent bifrontal craniotomy. The encephalocele was found to herniate through a defect in the left cribriform plate and communicate with the left frontal sinus. The encephalocele was resected and the cribriform plate was reconstructed with split-thickness autograft and titanium mesh. Exenteration and cranialization of the frontal sinus were performed bilaterally and the frontal sinus ostia were plugged with an abdominal fat graft. Both an external ventricular drain and lumbar drain were inserted to divert CSF while the repair healed. The postoperative course was uneventful with no signs of CSF leak and the patient was successfully discharged on postoperative day nine with no recurrence of symptoms. The patient remained well and symptom-free at the six-month follow-up.This is a rare case of a patient with a large primary  transethmoidal encephalocele that presented for the first time at 43 years of age. Despite the lack of craniofacial abnormalities, the absence of reported significant trauma history, lifelong duration of symptoms, and bony remodeling changes on imaging are highly suggestive of a congenital etiology. Additionally, no lesions suspicious for malignancy were present on imaging, which may also cause a secondary encephalocele .Those undiagnosed at birth can present with nasal obstruction, visual symptoms, or rhinorrhea ,3. If unAn interesting link exists between obesity, idiopathic intracranial hypertension, and spontaneous CSF leak of the anterior skull base which may result in an encephalocele -9. In thEndoscopic endonasal approaches have become the mainstay of treatment for CSF leaks and encephaloceles in the anterior skull base, with closure rates exceeding 90% -12. HoweIn conclusion, longstanding rhinorrhea accompanied by symptoms such as anosmia, headache, and visual abnormalities should prompt investigation into anterior skull base abnormalities. In the case of transethmoidal encephalocele, prompt surgical repair is essential to improve symptoms and prevent complications such as recurrent meningitis.


Cancer immunotherapies, including immune checkpoint inhibitors, elicit long-term clinical responses but many cancer patients do not respond. Intensive efforts are therefore underway to identify additional immune pathways that may be modulated to enhance the efficacy of existing immunotherapies. Bee venom strongly stimulates the immune system, and is used as a complementary therapy to treat cancer pain in patients with advanced tumors in China. Bee venom contains several allergenic protease inhibitors and peptides. It triggers hypersensitivity reactions; that is, it is an immune system agonist. The generation of a spontaneous T cell response against tumor-associated antigens requires innate immune activation; this drives type I interferon production. We report a patient with a relapsed and refractory liposarcoma who had undergone several operations, chemotherapies, and radiotherapies. The tumor was large. The patient had attained the maximum radiation exposure dose. The tumor was resistant to chemotherapy and was infiltrating the pericardium, lungs, and diaphragm. The patient was a poor candidate for resection. He thus received apitherapy (a combination of bee venom and acupuncture) to control pain; then apatinib (an anti-angiogenic drug) was given to inhibit tumor growth but was terminated early because the patient could not tolerate the side effects. Subsequently, a programmed death 1 inhibitor was combined with apitherapy. Bee venom served as an innate immune system agonist promoting immune cell priming and recruitment in the tumor microenvironment. The patient was finally able to undergo radical liposarcoma resection, and no evidence of recurrence was found at re-examination 16 months after surgery. Apitherapy, a therapy unique to traditional Chinese medicine, features both acupuncture and moxibustion (the burning and swelling caused by bee venom is similar to moxibustion). Acupuncture reduces cancer pain . This anDrugs that target programmed death 1 or programmed death 1 ligand 1 (PD-1/PD-L1) (immune checkpoint inhibitors) have become widely available in recent years , but fewThe patient was a 62-year-old man with a liposarcoma that had repeatedly relapsed. In February 2000, he underwent resection of a lipoma-like nodule on the left wall of the chest, but surgical pathology was not performed. In June 2004, he found a lump on the edge of the surgical scar. Three months later, a tumor was removed in the same hospital but again no tissue was sent for pathological examination. One month later, another mass developed on the left wall of the chest, and grew rapidly to attain dimensions of about 5 × 4 cm. He was admitted to a general hospital and underwent locally expanded, left wall excision on March 28, 2005. The surgical pathology indicated a pleomorphic liposarcoma with positive surgical margins. Then the patient underwent nine rounds of chemotherapy . Radiotherapy (30 Gy) was delivered to the surgical area of the left wall of the chest after chemotherapy concluded. The liposarcoma recurred once more (on October 30 2014) 5 cm below the left nipple. Four weeks later, he underwent a repeat, locally expanded excision of the same wall. A pleomorphic liposarcoma with positive surgical margins was again confirmed by surgical pathology (Apis cerana fabricius).On February 7, 2019, he experienced local pain and swelling on the left wall of the chest. The pain commenced as a pinprick, but then developed into a paroxysmal tear-like shape with a numerical rating scale (NRS) score of 6. The pain was not relieved by loxoprofen sodium tablets, and his sleep was severely affected. On April 8 2019 the patient was admitted to our oncology department for the first time. Positron emission tomography (PET)/computed tomography was performed early the following day and revealed a huge mass with irregular nodules and protrusions, of dimensions 10.5 × 5.4 × 7.2 cm, in the lower left wall of the chest. The boundary of the mass was unclear, and it was lobulated. The density was uneven, and local fat density shadows were evident. The average mass density was about 49.5 Hounsfield units. The mass exhibited enhanced fluorodeoxyglucose (FDG) metabolism, with an SUVmax of 15.07. The mass had invaded part of the pericardium, diaphragm, and interior of the left lung. The left fifth anterior rib was absent, and the cortex of the left sixth anterior rib was irregular. The FDG metabolism of the left sixth anterior rib was elevated, with an SUVmax of 3.2  Figure 5in situ hybridization at all times  amplification in tumor cells was detected by fluorescence ll times Figure 1ll times . MDM2 exll times , 10. Howll times . Immune ll times , 13. Somll times , 15. We ll times . As bee in vivo  for the publication of any potentially identifiable images or data included in this article.WY, DX, FH, and XX designed, organized, and supervised the study. WY, GY, YY, and HW drafted the manuscript. WY, JW, and DC analyzed the literature. DX and XX revised the manuscript. YZ, YG, JJ, and FH participated in the revision. All authors contributed to the article and approved the submitted version.The Medical and Health Research Project Funded by the Hangzhou Municipal Health and Family Planning Commission .The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


This report presents an extremely rare case of synchronous gastric cancer and primary adrenal diffuse large B-cell lymphoma (DLBCL). An 82-year-old man underwent computed tomography, which revealed a heterogeneous appearing and hypodense adrenal mass and a gastric mass with no enlarged lymph nodes in the neck, mediastinum, abdomen, and inguinal region. Upper gastrointestinal endoscopy revealed a protruding gastric tumor. The specimens obtained from endoscopic biopsy were histologically confirmed to be adenocarcinoma. The hormonal findings eliminated functional adrenal tumor. The patient underwent distal gastrectomy with regional lymph node resection for gastric cancer and incisional biopsy of the adrenal mass. Based on the pathological findings, diagnoses of mixed mucinous and tubular adenocarcinomas of the stomach and adrenal DLBCL were confirmed. Postoperation, the patient received rituximab combined with low-dose doxorubicin, cyclophosphamide, vincristine, and prednisone (R-miniCHOP). Six courses of R-miniCHOP were planned, but were completed in only one course at the patient’s request. The patient died 2 months after surgery. Gastric cancer is one of the most common gastrointestinal tract carcinomas. Alternatively, primary adrenal lymphoma is extremely rare, accounting for less than 1% of all non-Hodgkin’s lymphomas .Diffuse large B-cell lymphoma (DLBCL) is an aggressive lymphoma that accounts for about 30% of all cases of non-Hodgkin’s lymphoma . The synAn 82-year-old man was referred to our hospital for further investigation of a right adrenal mass and a gastric mass, which were coincidentally revealed by a follow-up computed tomography (CT) for an abdominal aortic aneurysm. The patient had a history of hypertension, diabetes mellitus, hyperlipidemia, cerebral infarction, and angina pectoris. The patient was an ex-smoker, consuming one pack per year for 55 years, and was a social drinker. In addition, the patient had a 6-kg weight loss in 1 month, abdominal pain, and anorexia. Physical examination showed no swelling of the superficial lymph nodes. The patient’s blood pressure was 139/68 mmHg, pulse was 65 beats per minute, and body temperature was 36.8 °C. The patient had Eastern Cooperative Oncology Group performance status of 1.Noncontrast-enhanced CT revealed a heterogeneous appearing and hypodense adrenal mass with irregular margins and shape, 70 mm in size, and a gastric mass, 30 mm in size, with no enlarged lymph nodes in the neck, mediastinum, abdomen, and inguinal region Fig. a, b. ConSurgical exploration of the abdomen revealed no evidence of ascites or metastasis to the liver or peritoneum. Distal gastrectomy with regional lymph node resection was performed for gastric cancer. The adrenal gland mass was tightly adherent to the surrounding tissues; therefore, the mass was judged to be impossible to resect, and an incisional biopsy was performed.Helicobacter pylori were detected by Giemsa staining in the resected specimens. The resection margins were free of tumor cells. The tumor had invaded the submucosal layer at a depth of 4 mm in the submucosa. The resected regional lymph nodes revealed no evidence of metastasis. The patient was diagnosed with T1bN0M0, stage IA according to the 8th edition of the Union for International Cancer Control TNM Classification of Malignant Tumors.Hematoxylin and eosin staining of the adrenal mass showed a diffuse proliferation of large and atypical lymphocytes accompanied by small lymphocytes Fig. a. ImmunoBone marrow aspiration indicated no bone marrow invasion of DLBCL. The patient had stage IE according to the Ann Arbor staging system. According to the National Comprehensive Cancer Network International Prognostic Index , two advGastric cancer is the fourth most common cancer and the second most common cause of cancer-related death worldwide . AlternaWith the development of diagnostic imaging, the number of adrenal tumors found by chance is increasing , 25. AmoThe adrenal gland is an organ with abundant blood flow and is prone to hematogenous metastasis from malignant tumors . AdrenalIn the present study, incisional biopsy of the adrenal mass was performed concurrently with gastrectomy for gastric cancer. The adrenal mass was diagnosed as a primary adrenal lymphoma, a disease with poor prognosis, and gastric cancer was pathologically diagnosed as stage IA. Through gastrectomy, chemotherapy for primary adrenal lymphoma had become less tolerable. The preoperative gastric cancer was not large in size and had no enlarged lymph nodes, and there was no tumor bleeding or gastrointestinal obstruction. The gastric cancer was of much less concern than the adrenal mass on imaging. Therefore, in the present case, the histopathological confirmation of the adrenal mass should have been performed first by diagnostic laparoscopy without gastrectomy. In this way, the diagnosis of the adrenal mass could have been made less invasive, and chemotherapy would not have been less tolerable by preserving the stomach. When gastric cancer and an adrenal mass are found simultaneously, the diagnostic approach of the adrenal mass needs to be carefully considered, taking into account the malignancy and progression of each tumor, and when gastric cancer is of much less concern than the adrenal mass as in this case, it is necessary to make a diagnosis of the adrenal mass first before treating the gastric cancer.Secondary involvement of the adrenal glands by a malignant lymphoma has been reported to occur in as many as 25% of patients with the disease . HoweverTable As this study was a single-patient case report, the accumulation of prospective evidence from more patients in multiple institutions should confirm the findings of this case report. Primary adrenal DLBCL are relatively rare; therefore, the number of patients treated in a single institution is limited. However, the current findings provide important information that can contribute to the development of a treatment strategy for the concomitant occurrence of gastric cancer and adrenal mass.In conclusion, this case report detailed the case of a patient with synchronous gastric cancer and primary lymphoma of right adrenal gland. This case indicates that in patients with concomitant gastric cancer and an adrenal mass, clinicians should be careful not to assume that the adrenal mass is an adrenal metastasis from gastric cancer without histopathological confirmation and need to consider the possibility of adrenal lymphoma. Furthermore, clinicians need to carefully consider the diagnostic approach of the adrenal mass, taking into consideration the malignancy and progression of each tumor.


Myopericytoma (MPC) is a rare, benign tumour often presenting as a cutaneous growth commonly in the lower extremities. It is distinguished by its concentric layering of spindle shaped myoid appearing cells perivascularly. These cells diagnostically stain positive to alpha smooth-muscle actin and rarely positive to desmin stain. This case study reviews the presentation of a 56-year-old male with a slow-growing, pre-tibial lesion developing over a 7–8 year period. This lesion was asymptomatic and demonstrated vascular involvement on ultrasound scan. This lesion measured 19 × 15 × 9 mm histologically and contained bland spindle cells surrounding vessels that interestingly stained positive to both alpha smooth-muscle actin and desmin. The histological findings in correlation to clinical presentation and imaging led to a diagnosis of MPC. Myopericytoma (MPC) is a rare, benign, slow-growing tumour that most commonly presents on the lower extremities . These lA 56-year-old male presented with a 7–8 year history of a slow-growing firm lump on his right distal tibia, midline on his anterior leg. The patient was otherwise in good health with no history of previous skin lesions and no family history of same. The lesion was subcutaneous and largely painless with no cutaneous manifestations. On physical examination, the nodule was 27 × 22 mm pre-tibial, 6 cm from the ankle joint. It was firm to palpation, subcutaneous with no skin discolouration or deformation. Ultrasound scan of the right lower leg demonstrated a well-defined, oval hypoechoic solid lesion with internal vascularity in the subcutaneous tissues measuring 18 × 7 × 20 mm. Biopsy was not performed due to vascularity of the lesion. Presumed diagnosis included fibroma, schwannoma or neurofibroma. The lesion was excised with clear margins. Microscopic examination revealed a well circumscribed, unencapsulated spindle cell lesion associated with varying sized dilated congested vessels, some of which showed a haemangiopericytomatous (HMP) pattern. The stroma included bland spindle cells seen surrounding blood vessels. No evidence of necrosis, atypia or mitotic activity was visualised. Further investigation with immunohistochemistry demonstrated spindle cells positive for both alpha smooth-muscle actin and desmin, with desmin showing variable positivity. The tumour cells were negative for CD-34. The examination, investigation and pathology are in keeping with a diagnosis of MPC. The lesion as stated above was completely excised, the wound demonstrated good recovery and no further surveillance required.MPC is a rare, cutaneous, benign lesion that has only recently been described in the medical literature. Its microscopically distinctive features include its perivascular concentric layering of stromal cells . MPCs coet al. in 1992 who noted myofibromatosis-like haemangiopericytomas in the thyroid of a 5-year-old boy [et al. proposed these cells are termed ‘myoperiocytes’ following review with electron microscope and immunohistochemical analysis [et al. further described myoperiocytoma after examining 24 subcutaneous tumours [et al. describe the distinguishing feature from myofibroma being the distinctive periluminal proliferation of round to oval cells lying in a concentric pattern [MPCs tumours were first described in a case study by Dictor  pattern .et al. reviewed 54 cases of MPCs and identified these lesions to most frequently present in the skin and soft tissues of lower extremities and occur more frequently in males than females in a wide age bracket [et al. identified only three cases positive to desmin stain [MPCs most commonly present as slow-growing painless firm lesions that are well circumscribed . Mentzel bracket . This isin stain . All casin stain . As statThis case demonstrates a classical presentation of the rare tumour, MPC, which was completely excised surgically with good wound recovery. Interestingly, this was a rare form of the tumour demonstrating uptake of desmin on immunohistochemistry staining in the pathology laboratory.


Hemolymphangioma is a congenital malformation of blood vessels and lymphatic vessels, commonly found in the head, neck, and subcutaneous, rarely in the viscera and extremely rarely in the liver. In this case, a 6-year-old boy was found to have abdominal distension for more than 2 months with no other obvious symptoms. Physical examination revealed a large abdominal mass that was hard and not mobile. Laboratory tests found no obvious abnormity. Preoperative ultrasound and CT showed a huge cystic and solid-cystic tumor in the abdomen with close relationship to the right lower margin of the liver and fluid accumulation in the abdominopelvic cavity. The preliminary diagnoses were a malignant tumor of embryonic origin and undifferentiated sarcoma. Liver tumor resection was performed in our hospital, and the postoperative pathology was diagnosed as hepatic hemolymphangioma. The patient recovered well after surgery. It is easy to diagnose a large abdominal mass in a child as a malignant tumor of the liver and delay the treatment—no obvious symptoms, no obvious abnormalities in laboratory tests, and imaging shows a multiocular cystic lesion with clear borders and no invasion of blood vessels, indicating that the possibility of this disease should be considered. The tumor has an abnormal rich blood supply, and preoperative imaging evaluation clearly shows the vascular pathway and blood supply status to help optimize the surgical plan. Hemolymphangioma is a congenital malformation of blood vessels and lymphatic vessels, which is a non-genuine tumor. Its occurrence in the liver is extremely rare, and only three cases –3 have bThe child was 6 years old, male, with abdominal distension found 2 months before admission, with no obvious precipitating factors and no abdominal pain or diarrhea. A large abdominal mass was palpated on physical examination, hard and without obvious mobility; the child's development was normal. Laboratory tests  did not show any significant abnormalities.Ultrasonography showed a huge multiocular cystic mass in the right lower abdomen with regular morphology, intact envelope, close relationship with the liver, uneven internal echogenicity, scattered anechoic areas within the hyperechoic zone, multiple hyperechoic light bands within the cyst wall, and abundant blood flow signal within the mass, with no obvious dilatation of portal vein and intrahepatic bile duct.3). No significant enhancement was seen in the cystic component; the mass was closely related to the right lower margin of the liver which showed a “cup-like” change, and branches of the intrinsic hepatic artery were seen in the arterial phase penetrating the mass  and the average red blood cell volume  was slightly reduced. Biochemical parameters showed that alanine aminotransferase  and γ-glutamyl transpeptidase  increased. It is recommended that patients be reviewed regularly after 1, 3, and 6 months later.The mass was cystic solid, about 10 × 15 × 16 cm, with intact peritoneum and regular borders; the cut surface was grayish white, grayish brown, medium in texture, locally multilocular cystic, containing yellowish green clear fluid. Microscopically, the tumor was seen to consist of abnormally dilated multilocular lymphatic vessels and blood vessels; hepatocytes and small bile ducts were seen inside . ImmunohUltrasound review at 3 weeks postoperatively showed that the operation area recovered well and no tumor residue was found. There was no abnormality in the physical examination and laboratory examination . After 3 months of follow-up, the child has good growth and development, no abnormal discomfort symptoms, and no abnormality in ultrasound and laboratory examination.Hemolymphangioma is a congenital malformation of the vascular and lymphatic system, the formation of which may result from obstruction of the venolymphatic communication between the dysembryoplastic vascular tissue and systemic circulation , 5.It is commonly found in loose connective tissues such as the head, neck, subcutaneous, etc. Its occurrence in the liver is extremely rare, and after searching , as of October 10, 2021, there were only three reports on hepatic hemolymphangioma . Among tThe clinical symptoms of abdominal hemolymphangioma are atypical, often presented with abdominal distension, abdominal pain, or palpable abdominal masses –9, and mAs a benign tumor, hemolymphangioma is treated by surgical resection, which has a good prognosis. However, its potential aggressiveness has been confirmed in the literature –13, and A large abdominal mass with abdominopelvic fluid is easy to be misdiagnosed as hepatoblastoma, undifferentiated embryonal sarcoma of the liver, and infantile hemangioendothelioma of the liver. Hepatoblastoma is usually located in the right lobe of the liver, grows exophytically, and usually presents as a large round-like or irregular lobulated mass with pseudo-envelope and well-defined borders; CT scan is mixed isointense or hypointense, which is always less dense than normal liver tissue, with internal hemorrhage, necrosis, cystic changes, and calcification. Enhancement scan is heterogeneous . UndiffeHemolymphangioma is extremely rare in the liver. It has no specific symptoms and no obvious abnormalities in laboratory tests, and the tumor is usually large. When it occurs in children, it can easily be misdiagnosed as a malignant tumor. If imaging shows a huge multilocular cystic lesion with mild progressive enhancement of the cyst wall, separation, and a solid portion, with clear borders, this needs to be considered a possibility of hemolymphangioma. As a benign tumor, hemolymphangioma is treated by surgical resection, which has a good prognosis. However, this tumor has an abnormally rich blood supply, and the adjacent large vessels are obviously compressed. Preoperative imaging evaluation clearly shows the vascular alignment and blood supply condition, which can help optimize the surgical plan.The original contributions presented in the study are included in the article/The present study was approved by the Ethics Committee of Lanzhou University Second Hospital . Written informed consent to participate in this study was provided by the participants' legal guardian/next of kin. Written informed consent was obtained from the individual(s), and minor(s)' legal guardian/next of kin, for the publication of any potentially identifiable images or data included in this article.YL: resources and writing original draft preparation. LT: software, investigation, and visualization. YX: formal analysis and data curation. JL: writing review and editing, supervision, project administration, and funding acquisition. All authors have read and agreed to the published version of the manuscript.This work was supported by the Lanzhou Talent Innovation and Entrepreneurship Project (Grant Number: 2020-RC-49) and the Basic Research Innovation Group Project of Gansu Province (Grant Number: 21JR7RA432).The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Pancreatic pseudocysts are a common complication of pancreatitis. However, mediastinal extension of a pseudocyst is rare and often presents with atypical symptoms. We present a case of mediastinal extension of a pancreatic pseudocyst in a 56-year-old woman with a history of alcohol-related chronic pancreatitis, who presented with acute on chronic epigastric abdominal pain and atypical chest pain. Serum lipase was elevated, and imaging by contrast-enhanced computed tomography (CT) demonstrated a paraesophageal fluid collection. This collection was continuous with a peripancreatic pseudocyst and extended into the posterior mediastinum via the esophageal hiatus. Mediastinal extension of a pancreatic pseudocyst was confirmed by magnetic resonance imaging (MRI). The patient was managed conservatively in the hospital with parenteral nutrition therapy, pain control, and close imaging observation. The patient was discharged home to continue conservative management and close imaging follow-up. An initial follow-up CT examination 8 weeks after discharge revealed interval decrease in the posterior mediastinal collection but also interval development of loculated left pleural and pericardial effusions. A fluid collection in the setting of pancreatitis may represent an abscess, hematoma, or pseudocyst. A pancreatic pseudocyst represents a localized peripancreatic amylase/lipase-rich homogenous fluid collection encapsulated by a fibrous wall, presenting greater than 4 weeks after a clinical episode of acute pancreatitis . MediasCross-sectional imaging (CT/MRI/MRCP) is essential in identifying a pseudocyst, ectopic extension of the fluid collection, and complications. MRI/MRCP will demonstrate a unilocular, encapsulated, homogenous fluid collection which exhibits high T2 and low T1 signal . BiocheManagement of mediastinal extension of a pseudocyst requires a multidisciplinary approach and depends on the clinical presentation, imaging findings (size and location), and local clinician expertise. Acute fluid collections after acute pancreatitis may spontaneously resolve in 50% of patients; however, persistent (greater than 4 weeks) fluid collections may develop into an abscess or a pseudocyst in the remaining 50% of patients [O'Connor et al. ]. On conMediastinal extension of a pancreatic pseudocyst is a rare complication of pancreatitis with an atypical presentation and potentially catastrophic complications. A high index of suspicion is required to prompt cross-sectional imaging and diagnosis. Treatment is difficult and requires a case-based multidisciplinary approach.


Isolated fallopian tube torsion (IFTT) is a rare cause of gynecological acute abdomen, is easily misdiagnosed and often has a delay in diagnosis. IFTT with paraovarian cysts is most frequently reported in studies. Here, we reported a patient diagnosed with IFTT associated with a paraovarian cyst, and we conducted a literature review for IFTT, aiming to identify valuable information that will be helpful for diagnosis and treatment for fallopian tube torsions.A 13-year-old girl presented with a 10-day history of right lower abdominal pain that worsened 2 days before presentation. On presentation, ultrasound showed a 5.8 * 5.5 cm hypoechoic cyst adjacent to the right ovary, and between the cyst and ovary, a tortuous thickened tube was visualized. Laparoscopy revealed a triple torsion of the right fallopian tube with a 6-cm paraovarian cyst, and tubal conservation surgery was performed. The postoperative course was uneventful. Histopathological diagnosis revealed serous papillary cystadenoma.Paraovarian cystic dilatation often occurs in adolescence and can induce fallopian torsion when the size of the cyst reaches 5-cm. In our review, the median age of patients diagnosed with IFTT with paraovarian cysts was 15 years old, and the main clinical manifestation was emergency abdominal pain. The associated symptoms were variable, and vomiting was the most commonly associated symptom. Salpingectomy was the most common procedure performed; however, timely surgical intervention can effectively avoid salpingectomy. IFTT is the torsion of the fallopian tube on its own axis. It is a rare cause of gynecological acute abdomen and was first reported by Bland-Sutton . The estA 13-year-old sexually inactive girl was admitted to our department due to a 10-day history of right lower abdominal pain, which worsened the last 2 days before presentation. It was suspected that she had an ovarian torsion and was treated by a local hospital with medication that did not relieve the symptoms. The physical examination suggested lower abdominal tenderness without defense. Pelvic examination revealed normal external genitalia. A cystic mass approximately 6-cm in diameter was palpable behind the uterus. Laboratory evaluations showed normal blood tests. Transabdominal and transrectal ultrasounds showed normal ovaries and a 5.8 * 5.5 cm hypoechoic cyst adjacent to the right ovary, and between the cyst and ovary, a tortuous thickened tube was visualized  was salpingectomy, and one of the procedures was a radical operation performed due to malignant paraovarian cancer found by rapid pathological examination. Conservative surgical management was performed in six out of 19 cases, in which the salpinges appeared viable after detorsion. Histopathologic diagnosis was reported in 17 cases; only six cases had definitive diagnoses, including three cases of mesothelial origin, one case of mesonephric origin and two cases of paramesonephric origin.Paraovarian or paratubal cysts are present in approximately 10% of all adnexal masses . They arIn general, parovarian cysts could be found at any age, and most of them were benign tumors. Paraovarian cysts are usually detected by ultrasonography or during surgery. Nevertheless, due to hormonal activity, especially in adolescence or during pregnancy, cystic dilatation often occurs . In thisPatients presented with abdominal pain, which could be described as gradual or acute, persistent or intermittent, and mild or severe. We found that 17 cases had the mode of pain recorded, and all of them presented with emergency abdominal pain, such as acute pain, severe pain, and aggravated pain. Some of the cases had a history of previous mild pain that subsequently worsened. Associated symptoms were variable and the most frequent symptom was vomiting (9/20), which was considered a stress response to the intense abdominal pain. On physical examination, mild tenderness was present in all patients, and peritoneal signs developed in advanced cases (if the condition progressed) . The medP = 0.651), demonstrating that necrosis was not associated with torsion times.The majority of fallopian tube torsions occurred on the left (14/20). The most likely explanation was that the sigmoid colon on the left provides cushioning as an accessory to prevent torsion by limiting the torsional activity, and patients with right abdominal pain more often underwent surgery because of the suspicion of appendicitis . FifteenUltrasonography was performed as the first diagnostic examination in all cases. The most specific characteristic of imaging of IFTT with paraovarian cysts was the presence of normal ovaries and a cystic mass, with or without a dilatated fallopian tube mass, which was due to tubal edema due to the torsion. The “break sign” was also another specific sign of IFTT, which showed narrowing at the extremity of the tube due to the torsion . Color DIn this study, most patients (13/19) underwent salpingectomy, and only six patients underwent more conservative surgical management, including detorsion of the tube and removal of paraovarian cysts. Surgical decisions depended on the observations of surgeons during the operation and whether the tubes developed normal circulation without necrosis after detorsion. Bertozzi et al.  reported


Pseudolipoma, also known as pseudolipoma of the Glisson's capsule, is an encapsulated lesion that contains degenerated fat and is enveloped by the liver capsule. In this report, we discuss a 37-year-old male presenting with dysuria and microscopic hematuria who revealed an incidental finding of a pseudolipoma on a CT scan of the abdomen. Pseudolipoma, also known as pseudolipoma of the Glisson's capsule, is a mass of fat tissue encapsulated and enveloped by the liver . It origA 37-year-old male presented due to dysuria and microscopic hematuria which was identified on urine analysis. Computed tomography of kidneys, ureters and bladder (CT KUB) showed no evidence of hydronephrosis or urolithiasis. An incidental cyst was identified within the liver dome (segment VIII), with flecks of calcification. Otherwise, the CT appearance of the liver parenchyma was normal with no other focal lesions Figure . CystoscAn MRI was done to further characterize the liver lesion, and this showed a cystic lesion which was isointense on T2 with complete signal drop out in the out of phase and fat saturated T1 images. The T1 hyperintensity is believed to be artifactual secondary to the calcification seen on CT Figure . The case was discussed at the upper gastrointestinal tract multidisciplinary team meeting, and the consensus was that this represents a benign pseudolipoma of Glisson's capsule.The liver consists of an outer serous layer, covering the majority of the liver, and an inner layer called the Glisson's capsule. The serous layer does not cover the bare area, porta hepatis, and the attachment site of the gallbladder to the liver. The Glisson's capsule, on the other hand, encapsulates the entire liver. This anatomic features along with the physiologic functions of the liver, exposes the liver capsule to a multitude of complications and disease processes . PseudolPseudolipomas are masses of degenerating fat that encapsulate and adhere to the liver, usually in the subcapsular space. They are thought to be the result of either receding hepatic lipomas or epiploic appendages . On pathOur patient is 37 years old and presented with dysuria and microscopic hematuria. Inflammatory markers and renal function were normal. CT KUB shows no hydronephrosis, however, an incidental cyst was identified within the liver dome (segment VIII), with flecks of calcification. Cystoscopy and CT urogram were normal and the patient’s symptom improved after a course of antibiotic.An MRI was done to know more about the nature of the liver lesion. The T1 hyperintensity is believed to be artifactual secondary to the calcification seen on CT. Diffusion weighted imaging showed no restriction diffusion while T2 showed a cystic lesion which was isointense on T2 with complete signal drop out in the out-of-phase and fat saturated T1 images.Further discussion by upper gastrointestinal tract multidisciplinary team meeting agreed that this represents a benign pseudo lipoma of Glisson's capsule.Pseudolipoma of Glisson's capsule are benign lesions composed of degenerating fat, likely derived from a dislodged epiploic appendage. Affected patients usually show no symptoms, however, abdominal pain similar to appendicitis or diverticulitis may occur. There is minimal evidence to show whether a patient's past histories of abdominal surgery are risk factors in the development of these lesions.


Lipofibromatous hamartoma (LFH) is a benign tumor that causes nerve enlargement due to fatty adipose tissue infiltration around bundles of peripheral nerves. It most commonly occurs at the median nerve with associated macrodactyly. We present an uncommon case of LFH that affected a digital branch of the median nerve without macrodactyly. Lipofibromatous hamartoma (LFH) is a benign tumor that causes nerve enlargement due to fatty adipose tissue infiltration around bundles of peripheral nerves. It most commonly occurs at the median nerve with associated macrodactyly. We present an uncommon case of LFH that affected a digital branch of the median nerve without macrodactyly. Its clinical presentation may vary, but its main symptoms are associated with neural compression, and the most commonly affected site of LFH is the median nerve. Its diagnosis can be confirmed after observing pertinent changes in ultrasonographic and magnetic resonance imaging (MRI), but when in doubt, a biopsy of the lesion is needed.2An 18‐year‐old woman without any medical condition or genetic disorder presented after a fall from a horse. She had forced traction of the middle finger of the left hand from the reins of the horse.The patient presented with insidious burning pain and numbness in the dorsal part of the proximal interphalangeal joint of the middle finger. No fractures were seen on radiographs; therefore, analgesic treatment was provided. After 4 months, pain remained constant and worsened with direct pressure of the proximal interphalangeal joint. She denied having fever or weight loss.Physical examination revealed a diffuse mass (approximately 1 × 1 cm) on the ulnar and dorsal side of the proximal phalanx of the middle finger of the left hand. The mass was not adherent to the skin and was mobile , the patient had no pain at the surgical site. The range of motion of the finger was preserved. She had no paresthesias but continued to have numbness on the dorsal part of the proximal interphalangeal joint of the middle finger.3Lipofibromatous hamartoma is an uncommon tumor that affects the peripheral nerves wherein fibroadipose tissue infiltrates the epineurium and perineurium.In 1953, MasonThe cause of abnormal growth of the nerve remains unknown.There are no pathognomonic findings for LFH.The differential diagnoses of LFH include ganglionic cysts, hereditary hypertrophic neuritis of Dejerine‐Sottas syndrome, vascular malformations, traumatic neuromas, schwannomas, neurofibromas, intraneural lipomas, fibromatosis, and plexiform neurofibroma.Radiographs may only be helpful in viewing macrodactyly changes and soft tissue opacity.In LFH, diffuse involvement of the nerve showing relative bland changes is consistent with a hamartomatous rather than a neoplastic tumor. The lesion is described as a hamartoma because of the overgrowth of connective tissue constituents observed. The proportion of fibrous tissue to fat vary from case to case.Due to the context of a benign tumor, there are different recommendations for the treatment of LFH. These include observation, biopsy, intraneural neurolysis, and/or carpal tunnel release.According to Mohamed et al4In conclusion, LFH is a rare condition with different clinical presentations. Evidence suggests that the diagnosis can be made with ultrasound and MRI without the need for a biopsy. In asymptomatic patients, treatment can be limited; however, if the tumor is symptomatic, surgical excision can be performed. In our case, the LFH was symptomatic, presented in an atypical location, and was not associated with macrodactyly; therefore, we decided to perform a complete excision. This pathology still needs further research to better understand its pathophysiology and to determine the best treatment option for it based on patient characteristics.None declared.JBS, RBF, and RASM were involved in patient care and wrote and reviewed the manuscript. LECC and LMBL reviewed the pathological images and provided histopathological research. All authors were involved in the editing and final approval of the manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor in Chief of this journal.


Small bowel obstruction (SBO) is a common condition requiring urgent attention that may involve surgical treatment. Imaging is essential for the diagnosis and characterization of SBO because the clinical presentation and results of laboratory tests may be nonspecific. Ultrasound is an excellent initial imaging modality for assisting physicians in the rapid and accurate diagnosis of a variety of pathologies to expedite management. In the case of SBO diagnosis, ultrasound has an overall sensitivity of 92% (95% CI: 89–95%) and specificity of 93% (95% CI: 85–97%); the aim of this review is to examine the criteria for the diagnosis of SBO by ultrasound, which can be divided into diagnostic and staging criteria. The diagnostic criteria include the presence of dilated loops and abnormal peristalsis, while the staging criteria are represented by parietal and valvulae conniventes alterations and by the presence of free extraluminal fluid. Ultrasound has reasonably high accuracy compared to computed tomography (CT) scanning and may substantially decrease the time to diagnosis; moreover, ultrasound is also widely used in the monitoring and follow-up of patients undergoing conservative treatment, allowing the assessment of loop distension and the resumption of peristalsis. Small bowel obstruction is a common disease; its incidence in patients who present to the emergency department (ED) is estimated at 2–8%, and about 15% of these patients are admitted to the surgical unit . The ter•Confirm or exclude intestinal obstruction ;•Define the level of obstruction ;•Define the cause of the obstruction;•Define the severity of the obstruction (staging).Imaging should achieve the following specific aims:The imaging methods used in the evaluation of a patient with suspected bowel obstruction are conventional radiology, ultrasound, computed tomography (CT), and magnetic resonance imaging (MRI). CT is the gold-standard imaging modality in the diagnosis and staging of small bowel obstruction ,22,23,24Global View technique ..12].Once the obstructive fulcrum has been identified, it is advisable to try to find the cause of the obstruction, easier in the case of a hernia or extraluminal mass , but morThere are some interesting tips and tricks that can help in the ultrasound evaluation of SBO ,12,35:Bypass technique: in the case of intestinal obstruction, with the presence of bowel gas distension especially at an early stage of the disease, it may help to bypass the intestinal air by placing the ultrasound probe more laterally to highlight fluid stasis.Graded compression technique: it may be useful to use graduated compression with a probe in order to improve the quality of the images; this technique is largely used in ultrasound bowel investigation. Point of pain: after completing the ultrasound examination with the convex probe, it is very important to ask the patient to locate the point of greatest pain. Through a clinical ultrasound evaluation, guided by the patient’s symptoms, it can be possible to identify the fulcrum and the cause of obstruction.Ultrasound criteria for the diagnosis of SBO can be divided into diagnostic and staging criteria  23]. . 23]. The diagnostic criteria include the presence of dilated loops and abnormal peristalsis while staging criteria are represented by parietal and valvulae conniventes alterations and by the presence of free extraluminal fluid.Small bowel dilatation is defined as bowel diameter ≥2.5 cm measured from outer wall to outer wall ,35,37. APeristalsis alteration represents a fundamental criterion for the diagnosis of mechanical ileus . In an eThe persistence of obstruction causes an increase in endoluminal pressure, and the liquid content normally present in the intestinal lumen cannot be reabsorbed. Bowel layers act as a sponge, determining the passage of fluid in the peritoneal cavity a,b. In tParietal changes are characterized by the presence or absence of parietal and valvulae conniventes thickening and parietal wall stratification. The evaluation of parietal changes follows a dichotomic diagnostic process based on the reference values : thickened walls/valvulae conniventes (YES/NO) or thinned walls (YES/NO) a,b 29,3,35. AlthThe valvulae conniventes  are permanent folds composed of mucosa and submucosa that project into the intestinal lumen and are clearly visible in the case of fluid distension (keyboard sign) . At an eAncillary signs are useful in increasing diagnostic confidence.These refer to the ‘bowel loop jump’—the visualization of two groups of loops with a clear difference in size  or peristalsis (‘kinesis jump’). In both cases, a comparison should be made between the loop upstream and that downstream of the obstruction .Ultrasound is a highly accurate imaging method for the diagnosis and staging of mechanical ileus of the small bowel, and the ultrasound findings fully reflect the evolutionary patterns of the disease. Moreover, ultrasound can be used to evaluate intestinal peristalsis in real-time, playing a key role in diagnosis and patient monitoring.In addition, a patient with an ultrasound diagnosis of mechanical ileus occlusion can and should be admitted to the surgical ward.Currently, contrast-enhanced ultrasound (CEUS) for the assessment of loop vascular distress is still considered level II imaging and is not widely used since it is not always able to detect the loop immediately upstream of the obstructive fulcrum, which is the subject of ultrasonographic study ,45. Ultr


Retroperitoneal paragangliomas are rare tumors, they arise from ganglia along the sympathetic and parasympathetic chain. We report a rare case of a non functional paraganglioma in whom surgical resection was performed.A 35 years-old female presented with chronic abdominal pain, A contrast magnetic resonance imaging (MRI) of abdomen showed a well-defined Left latero-aortic cystic retro-peritoneal surgical resection using laparotomy was performed, The patiente recovered well and was discharged three days after surgery. Histological examination and immunohistochemical revealed a retroperitoneal paraganglioma.Non-functioning retroperitoneal paragangliomas are rare and are most often Isolated. Radiological techniques including, Contrast-enhanced computed tomography (CT) and Magnetic resonance imaging (MRI) are useful for identifying and locating retroperitoneal paragangliomas. surgical excision is still the most effective treatment when it possible. •We report a case of non-functional retroperitoneal paraganglioma.•Non-functional retroperitoneal paraganglioma is extremely rare and mostly benign.•A difficult tumour to diagnose and treat.•Complete surgical excision is the treatment of choice for extra-adrenal paragangliomas. Most of them are functional, with symptoms and signs of catecholamine overproduction, similar to pheochromocytoma except for the variation in the anatomic location The risk of malignancy may be as high as 20%–30% compared with a 10% risk of malignancy for adrenal pheochromocytoma [We report a rare case of a non functional paraganglioma in whom surgical resection was performed. This work has been reported in line with the SCARE criteria .2A 35 years-old female with a medical history of chronic abdominal pain for about four months was admitted to our hospital Abdominal examination was normal.Abdominal and pelvis Ultrasound (US) revealed a Left retro peritoneal formation measuring 4cm rounded well-defined with heterogeneous non-vascularized content. A contrast magnetic resonance imaging (MRI) of abdomen showed a well-defined Left latero-aortic cystic retro-peritoneal mass measuring 45 × 50 mm appear as hypointense in T1w and hyperintense in T2w sequence with multiple septa . Biochem3Paraganglioma (PG), also known as extra-adrenal pheochromocytoma, is a chromaffin cell tumor located at various sites along the sympathetic and parasympathetic chain, they can thus be found in the head, neck, thorax, abdomen, and pelvis. Retroperitoneal paragangliomas are rare tumors. Non-functioning retroperitoneal forms are even more rare and are most often isolated .Histologically, most are composed of well-differentiated neuroendocrine cells disposed in small clusters  or cords separated by prominent fibrovascular stroma. Distinctive within the cells in most tumors are dark neurosecretory granules that contain catecholamines. Sometimes the cells are spindle shaped. Mitoses are usually infrequent, but occasional tumors are overtly anaplastic and pleomorphic and contain numerous mitoses. The malignant potential is determined by local invasion and also distant metastases .Functional PGs can be diagnosed based on clinical presentation, the most common type of presenting symptom were associated with catecholamine-secreting paragangliomas: Episodic headache, diaphoresis and tachycardia. Episodic hypertension has been identified as a characteristic feature of catecholamine-secreting paragangliomas and is used for the differential diagnosis of paragangliomas, laboratory examination showing elevated catecholamines and their metabolites (VMA) in the blood and urine. Nonfunctional PGLs are mostly found incidentally or through symptoms caused by compression of the surrounding organs . In our Radiological techniques including Ultrasound, Contrast-enhanced computed tomography (CT) and Magnetic resonance imaging (MRI) are useful for identifying and locating retroperitoneal paragangliomas, the imaging characteristics of retroperitoneal paragangliomas included soft-tissue masses in the sympathetic chains associated with the abdominal aorta, cystic degeneration and necrosis inside the masses functional imaging, such as PET scanning and MIBG scintigraphy may help detect primary or metastatic tumors that could be missed on CT/MRI , in our A complete surgical resection is still the most effective and the only potentially curative treatment and should be attempt even in patients with distant metastasis.Open exploration and resection is the standard surgical management of PG, however, laparoscopic resection of PG is considered challenging because of the altered anatomic location, dense peritumoral adhesions, high vascularity envelope, proximity to major blood vessels ,7. If a Patient with metastatic disease will require adjuvant radiotherapy while chemotherapy is restricted to patients not accessible for surgery and resistant to radionuclide therapy .4Non-functional retroperitoneal paragangliomas are rare group of tumors which are difficult to diagnose, owing to their silent and asymptomatic behavior, Once the diagnosis of a retroperitoneal paraganglioma has been made, surgical excision is required and is still the only curative approach.I declare on my honor that the ethical approval has been exempted by my establishment.None.Amal Hajri: writing the paper.Ahmed Ballati: Corresponding author writing the paper.Zakaria Essaidi: writing the paper.Driss Errguibi: study concept.Rachid Boufettal: study concept.Saad Rifki El Jai: correction of the paper.Farid Chehab: correction of the paper.Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient.Not applicable.DOCTEUR AHMED BALLATI.None.Not commissioned, externally peer-reviewed.The authors declare having no conflicts of interest for this article.


Symptomatic pituitary metastasis is rare; furthermore, it can result in diabetes insipidus and panhypopituitarism. Since diabetes insipidus is masked by concurrent panhypopituitarism, it can impede the diagnosis of pituitary dysfunction.A 68-year-old Japanese female suffering from pituitary and thalamic metastases caused by untreated breast cancer, underwent a biopsy targeting the thalamus, not the pituitary. She lacked prebiopsy pituitary dysfunction symptoms; however, these symptoms unexpectedly occurred after biopsy. Diabetes insipidus was masked by corticosteroid insufficiency, and she showed normal urinary output and plasma sodium levels. Upon commencement of glucocorticoid replacement therapy, the symptoms of diabetes insipidus appeared.In this case, thalamic biopsy, as opposed to pituitary biopsy, was performed to preserve pituitary function. However, pituitary dysfunction could not be avoided. Caution is necessary for asymptomatic patients with pituitary metastases as invasive interventions, such as surgery, may induce pituitary dysfunction. Moreover, with respect to masked diabetes insipidus, there is a need to carefully consider pituitary dysfunction to avoid misdiagnosis and delayed treatment. Pituitary metastases are rare, and are frequently asymptomatic or present with nonspecific symptoms . PatientThis is a case of pituitary metastasis accompanied by thalamus metastasis, with breast cancer as the source. The patient underwent a biopsy targeting the thalamus, with no prebiopsy DI symptoms. We opted not to biopsy the pituitary metastases to avoid pituitary dysfunction. After the biopsy, there was transient polyuria, which improved within a few days. The patient presented with continuous lethargy and fatigue. We inferred that it was induced by the progression of metastasis and peritumoral edema, and glucocorticoids were administrated accordingly. However, DI symptoms appeared upon glucocorticoid replacement therapy. This suggested that thalamic biopsy induced pituitary dysfunction, resulting in DI and panhypopituitarism. We concluded that the continuous lethargy and fatigue were signs of panhypopituitarism as opposed to brain edema. Moreover, DI was masked by ACTH deficiency, which led to normal urinary output and plasma sodium levels. We report an illustrative case of pituitary metastasis showing masked DI.A 68-year-old Japanese woman presented to our hospital with a skilled movement disorder of the left hand, which had gradually progressed over the previous few months. She showed normal consciousness and slight fatigue without thirst. She had no psychological problems and no challenges regarding financial, language, and cultural matters. She previously underwent a uterine myoma surgery and had two sisters who had been diagnosed with breast cancer. Brain magnetic resonance imaging (MRI) revealed two mass lesions. One lesion was 21 × 21 × 17 mm and was located in the right thalamus with accompanying brain edema. On T2-weighted imaging (T2WI), this was shown as high-density areas that progressed to the right pedunculus cerebri. The other lesion was located in the hypothalamus, infundibular stalk, and pituitary posterior lobe, which compose the hypothalamic–pituitary axis (HPA). The stalk was thickened on T1-weighted imaging (T1WI), showing an absence of the normal posterior pituitary bright spot (PPBS). Contrast-enhanced T1WI showed intense enhancement along the lesion edges and uniform enhancement within the lesions  were within the normal range. She underwent a stereotactic biopsy targeting the thalamus, but not HPA, lesions  0, her urine volume was > 3000 ml. At POD 1, plasma sodium levels increased to 155 mEq/l Fig. A, B. We Regular desmopressin administration ameliorated the polyuria Fig. A, C. IntThis is a case of thalamic and pituitary metastasis from breast cancer with accompanying central DI and panhypopituitarism, from which two learning points can be derived. First, an invasive intervention, specifically stereotactic biopsy in this study, may induce pituitary dysfunction. Second, central DI is masked by comorbid ACTH insufficiency.Numerous reports have indicated that metastasis to the pituitary gland is rare , 2, 4, 5In this case, thalamic metastasis, as opposed to pituitary metastasis or the primary malignancy, caused the primary manifestation and chief complaint . The pituitary metastasis was asymptomatic before the biopsy surgery. As postoperative DI has been observed in 10.2% of endoscopic transsphenoidal surgery , the tha2 receptor, adrenal insufficiency may amplify the ADH effects [Another factor that delayed diagnosis and treatment in this case was masked DI. Central DI results from deficient ADH synthesis or secretion, and its most common causes of central DI are benign or malignant neoplasms of the HPA . Patient effects .In this study, upon initiation of glucocorticoid replacement therapy, DI symptoms, such as polyuria, appeared. Therefore, the normal plasma sodium levels and daily urinary output observed until glucocorticoid replacement were due to masked DI. Glucocorticoid replacement is effective for the diagnosis of masked DI because glucocorticoid replacement increases the sensitivity for diagnosis in cases of pituitary metastasis . The fatAnother case report of pituitary metastasis discussed severe hyponatremia, which was induced by secondary adrenal insufficiency accompanied by DI . In thisPrevious studies have reported that most pituitary metastases occur in the posterior lobe , 2. ThisThere are no standardized treatment guidelines for the management of pituitary metastases . AlthougThe mean survival period after detecting pituitary metastasis is 10–14 months . In caseIn pituitary metastasis, it is important to consider that invasive examination or intervention may induce pituitary dysfunction, including DI or panhypopituitarism. Since ACTH insufficiency can mask DI, we must consider pituitary dysfunction even when urinary output and plasma sodium levels are normal. Careful monitoring of symptoms, including lethargy or fatigue, should be done to avoid delayed treatment.


Brunner's gland adenoma is a rare benign tumor of small bowel, often incidentally discovered during endoscopy or radiological imaging. Mostly they are asymptomatic or often present with nonspecific symptoms such as nausea, vomiting, gastrointestinal hemorrhage, iron deficiency anemia. We reported a 76 years old male case presented with chief complaints of vomiting and black tarry stool. General physical examination was normal except mild tenderness over epigastrium. Esophagogastroduodenoscopy revealed a pedunculated polypoid tubular structure with blind end distally of length approximately 10-12* 3.5*1.5 cm in the second section of the duodenum with multiple skipped ulcers on the exposed surface of it. Additionally, there were few erosions in the duodenum proximally and multiple superficial ulcerations in the antrum, associated with helicobacter pylori confirmed by rapid urease test kit. Surgical or endoscopic excision is the treatment of choice. We consider our case is the eldest case among Brunner's gland adenoma case in literature. Brunner's glands account 10% of benign neoplasm of the duodenum, the first described by Brunner in 16884 Mostly they are asymptomatic or present with chronic abdominal pain, nausea, vomiting, gastrointestinal bleedings, and anemia.5Generally, it is incidentally diagnosed during upper gastrointestinal endoscopy or sometimes computed tomography and magnetic resonance imaging as a tubular or cystic lesion in the duodenum.We are reporting a rare case and eldest case of BGA presented with upper gastrointestinal bleeding.76 years old male patient visited on the outpatient department with chief complaints of vomiting of blackish color contents and melena for 2 days. He had chronic episodic upper central abdominal pain for many years subsided by antisecretory medications; however, no history of melena previously. There was no significant previous medical and surgical history or investigation done such as endoscopy. On presentation, he was hemodynamically stable with normal vitals and biochemistry and hemoglobin was 11.9 gm/dl; however, stool for occult blood was positive and black tarry stool on per rectal examination. Therefore, he was admitted and pantoprazole 80 mg stat, then followed by 8mg/ hr via infusion pump given along with oral sucralfate. We performed an esophagogastroduodenoscopy on day second of admission, showed pedunculated and elongated tubular blind end distally polypoid mass with an approximate length of~10-12 cm, broad base 3.5*1.5 cm size, arising about the same level of the major papillary opening in second part of the duodenum . There w6 The exact pathophysiology of the Brunner's gland adenoma remains obscure, however, there are many postulations regarding the etiology of BGH. Increased gastric acid secretion is believed to be the cause of Brunner's gland hyperplasia as Brunner's gland maintains the alkaline condition in the duodenum, and also an association of BGH and achlorhydria; but Spellberg et al. didn't found regression of lesion with anti-acid secretary medication. Furthermore, Helicobacter pylori infection may play a major role in Brunner's gland hyperplasia or adenoma. It has been reported that there was the association of HP infection and BGA in 71% of cases,7 however, there was no HP association in both cases in another case report. 6 Above all, the pathogenesis remains duodenal dysembryoplastic lesion or hamartoma.Brunner's gland adenoma or Brunneroma is an extremely rare benign small bowel tumor arising from the Brunner's gland commonly in the second part of the duodenum. It is also known as Brunner's gland hamartoma. Although it is a rare variant with the incidence of <1 percent, there are many cases reports published in the literature.9 Those who presented with melena had larger size with maximum size reported up to 12cm,5 in our case also length was approximately 10-12 cm which is longer as previously reported.9 Additionally, age of the patient was older (76 years) than previously reported of 73 years old.1Generally, Brunner's gland adenoma is asymptomatic or presents with nonspecific symptoms with abdominal discomfort, nausea, vomiting, gastrointestinal bleeding, intussusception, or iron deficiency anemia. Recent studies reported melena as the presenting complaints as in our case although he had chronic episodic abdominal discomfort for 2 years.10 According to a recent study, dysplastic changes noted in 2.1% and invasive carcinoma in 0.3% of all Brunner's gland hyperplasia.10 Surgical resection is the gold standard especially for giant Brunner's gland hamartoma and unresectable mass; however, recent studies have prevailed endosco-pic resection of the large adenoma also have been safely done without complications. 5Brunner's gland adenoma commonly presented as hyperplastic or hamartomatous mucosal changes are always benign; however, rarely foci of metaplasia to malignant foci have been reported.In conclusion, although Brunner's gland adenoma have a very low incidence, recently more cases are reported in the literature, they can present as gastrointestinal bleeding, and diagnosed by endoscopic or radiological imaging. Surgical resection or endoscopic resection is equally possible in recent days.


Kimura disease (KD) is a chronic, inflammatory, benign disorder endemic to Asia that typically manifests as a triad of painless masses in the head and neck region, elevated eosinophils and serum immunoglobulin. It usually affects young men in their second and third decades of life and is rarely seen outside of the orient. This is a report of a case of KD in a young man of African descent who presented with a cheek mass. KD was not included in our differential diagnosis, and this report highlights the need to consider this entity, which can be easily missed due to its rarity in the Western world. There is no cure for the disease, and management includes medical and surgical modalities, but local recurrence or relapse is not uncommon. Kimura disease (KD) is a rare benign, chronic, inflammatory disorder for which the aetiology is largely unknown . It mostA 29-year-old male of African descent presented to the Plastic and Reconstructive Surgery Outpatient Department at a hospital, with a 3-year history of a non-tender, slow-growing swelling to the right cheek. He reported no constitutional symptoms, and his previous medical history was unremarkable.On examination, there was a 3.0 × 3.0 cm spherical, firm, smooth, non-tender mass on the right cheek. Clinically, the mass appeared confined to the subcutaneous plane with no involvement of the overlying skin or mucosa, and there was no associated lymphadenopathy.9/l with an eosinophil rate of 17.4%  and normal renal function.Ultrasound examination revealed a heterogenous lesion measuring 2.9 × 2.3 × 1.4 cm, with associated non-specific ipsilateral Level II cervical lymph nodes . HaematoAn excisional biopsy was performed, and the histopathological findings were those of numerous, variably sized lymphoid follicles set within a background of hyalinized fibrous and adipose tissues as well as germinal centres bordered by well-defined mantle zones and numerous eosinophils located in the inter-follicular areas, all in keeping with a diagnosis of KD . The resHe re-presented 2 years later with a larger 5.0 × 5.0 cm swelling in the same area of the right check . UltrasoHe is now 14 months postoperative since re-excision and has shown no signs of recurrence. He is being seen in the outpatient department on a 3-monthly basis.KD was first reported by Kimm and Szeto in 1937 in China as ‘eosinophilic hyperplastic lymphogranuloma’, but the details of the pathological features of ‘unusual granulation combined with hyperplastic changes in lymphoid tissues’ were subsequently described by Kimura in 1948 in Japan , 6. ThisClassical investigation includes the use of ultrasonography, computed tomography or magnetic resonance imaging to delineate the anatomical structures as well as to rule out more sinister conditions, e.g. tuberculosis, lymphoma, salivary gland cancers, all of which may mimic KD.In addition, these studies can facilitate image-guided biopsy of the lesion or associated lymph nodes in equivocal cases where tissue histopathology is essential in making the diagnosis , 6.Histologically, KD is identified by the presence of intact lymph node architecture with reactive and prominent germinal centres with dense eosinophilic infiltration of the interfollicular areas along with lysis of the follicles—all of which were present in this case . Tissue There is no universally accepted standard of management of KD, and there is no cure. Various treatment modalities have been described and include medical management, surgical intervention, radiotherapy and chemotherapy .Medical management mainly consists of continuous low-dose steroids and, less commonly, the use of immune modulators . HoweverSurgical excision alone is associated with recurrence rates of up to 40%, due to the ill-defined borders of these infiltrative lesions. Furthermore, given the benign nature of the disease, there is always the balance between the removal of involved tissue against the aesthetic and functional deficits, which may result from wide local excision .The use of radiation therapy alone has been shown to have local recurrence rates of up to 75%; however, when used as an adjunct to surgery, recurrence rates are significantly less when compared with either treatment modality as a single entity, and it is now generally accepted that surgery followed by low dose radiation offers the longest periods of remission , 8.KD is a rare entity in the Western world and particularly among persons of African descent, hereby, making the diagnosis exceedingly difficult in our setting.None declared.None.


Hairy polyps are benign embryological tumors of the head and neck region that are derived from two germinal layers, the ectoderm and mesoderm. At an incidence of 1 : 40000, hairy polyps are considered rare growths. Presenting symptoms of hairy polyps can vary greatly depending on the size and location of the tumor. To the best of our knowledge, our reported case is the first in the literature to highlight an extremely rare occurrence of two separate hairy polyps being simultaneously present in one patient, in the clivus and the nasion, with the presence of meningothelial cells within both tumors, histopathologically. With surgical resection as the management of choice, the approach of surgery differs greatly depending on many factors. Since the patient in our reported case had a cleft palate, we used a combined transnasal and transoral approach to fully release the clival mass and a direct skin incision for the nasion mass. Recurrence after complete surgical resection is rare, and if happens, it should rise suspicion of histopathologically misdiagnosed tumor. We amplified the importance of advanced radiological investigations along with proper multidisciplinary teamwork to exclude CNS connections and other histologically malignant tumors and to early pick up a possible simultaneous lesion. Hairy polyps are benign growths that originate from two germinal layers, the ectoderm and mesoderm. Being first described by Brown-Kelly in 1918, hairy polyps are embryological malformations that usually present in the early childhood period. Hairy polyp is a rare tumor in the head and neck, with an incidence of 1 case in every 40,000 individuals. We reported a unique case of two separate hairy polyps with meningothelial cells occurring simultaneously in one patient.15-month-old male baby, a product of an uneventful dizygotic pregnancy with his twin brother being totally normal, was delivered by cesarean section due to obstetric reasons. At the age of 7 days, he was admitted in another hospital for the excision of a tongue dermoid cyst prior to referral to our center. The patient was referred to KFSHRC primarily for the evaluation of an incomplete cleft palate with a tongue mass. On examining the patient, a cleft palate was noted with a midline hard tumor at the posterior edge of a bifid palate at a size of around 2.5 × 1.5 cm, along with a residual tongue lesion measuring 2 × 1.5 cm at the anterior dorsal surface of the tongue.The patient was planned to have MRI  that shoOn the next visit, CT facial bones were done and showed cleft lip and palate with other findings, as shown in The impression, based on radiological findings and clinical examination, was a skull base mass at the clivus, extending to the sphenoid sinus. In addition to that, the patient had a midline nasal mass in the area of the nasion with extension to the skin but without any sinus.Given that the patient had a cleft palate, the decision was made to perform combined endoscopic transnasal and transoral resection of the clival lesion with resection of the midline nasal structure. Informed consent for the surgery was then taken from the family.The patient was taken to the operation room. Nasal examination under general anesthesia using a 30-degree scope was done for the first time, and we were able to identify a clival hard lesion that was extending through the cleft into the oral cavity. Access to both sides of the lesion was achieved by a small posterior septectomy, lateralizing the inferior turbinate and incising the posterior septum. Using a diamond drill, the lesion was drilled off the clivus. And by using a true cutting instrument, all soft tissue attachments were released intranasally, superiorly, medially, and laterally. The remaining attachment was an anterior attachment to the floor of the nose, which was released by a transoral approach. At this stage, en block resection of the mass was achieved.For the other mass over the nasion, a midline incision was done over the nasion. After elevating the periosteum, a cyst was identified with resultant cavitation in the bone. The cyst was dissected completely, and the cavity was drilled using a diamond drill to remove any remaining soft tissue. The incision was then closed in two layers.Postoperatively, the patient was doing fine and tolerating oral feeding. After 24 hours, he was discharged with a clinic appointment.On histopathological examination, grossly, the clival mass was white-tan irregular firm tissue measuring 2 × 2 × 1 cm, with homogeneous white nodular cut surface. The midline nasal mass was white-tan hemorrhagic fibrous tissue, measuring 2 × 2 × 0.3 cm. So, both masses showed the presence of teratoid hairy polyps with meningothelial cells and tooth tissue noted. No endodermal elements were seen on the submitted specimen, as shown in Regular follow-ups with CT scans showed no progression in the teratoid over a period of 18 months postoperatively. Given that he is asymptomatic with no clinical signs of recurrence, the decision was elected to observe the patient on serial imaging keeping in mind that the patient may need surgical intervention through an endoscopic or craniotomy approach in the future depending on the symptoms and any progression. The parents were counselled in detail regarding their child's condition.Oropharyngeal and nasopharyngeal hairy polyps, although not common, but are well described embryological malformations in the literature. The first case of hairy polyp was reported by Brown-Kelly in the early twentieth century, 1918 . Hairy pHairy polyps of the oronasopharynx are benign growths that may present with a wide range of symptoms, depending on size and location. In our case report, we amplify the importance of advanced radiological investigations along with proper multidisciplinary teamwork to exclude CNS connections and to early pick up a possible simultaneous lesion.


Hemangiomas are benign vascular lesions frequently observed in infancy and childhood. A 14-year-old boy was referred to the hospital with a left-sided ear canal mass and hearing impairment. Otoscopic examination revealed a mass that occluded the canal. The transcanal surgical excision was performed. The mass was dissected from the skin, no bony erosion was noted intraoperatively, and the skin was returned back properly. The histopathological report demonstrated a capillary hemangioma of the external auditory canal.Hemangiomas are relatively common in the head and neck, but rarely detected in the external ear canal and tympanic membrane. Hemangioma of the ear canal may be asymptomatic and accidentally observed. Despite the rarity of the mass, it is important to consider them in the differential diagnosis of external auditory canal masses. The computed tomography and magnetic resonance imaging with gadolinium help to reveal the vascular nature of the mass. Complete surgical excision is the treatment of choice. Hemangiomas are recognized as benign vascular lesions in infancy and childhood mostly observed in head and neck areas. One-third of these tumors may present at birth. Hemangiomas of the external auditory canal and eardrum are rare conditions. It initially presents as a small vascular lesion in the deep posterior bony external auditory canal or at the posterosuperior tympanic membrane. It is usually asymptomatic till becomes bigger or bleeds due to the manipulation by the patient. Hemangiomas are classified into different histological categories: cavernous, capillary, and mixed. So far, only a few cases of capillary hemangiomas of the external auditory canal have been reported ,2.A 14-year old adolescent boy was referred to our otolaryngology clinic with a complaint of right-sided ear canal painless mass and hearing impairment. Based on the patient's statement, he experienced progressive hearing loss for one month to the extent that he had difficulty hearing. He also stated that the mass which was small at first grew fast and filled the ear canal. He denied any history of ear discharge, pain, tinnitus, and vertigo. According to the patient, the earlier attempts for mass excision were followed by the recurrence of the problem after a few weeks. He did not mention any other disease or drugs administration in his medical history. Moreover, no benign or malignant cysts or tumors were reported in his family members.On otoendoscopic examination, a broad base mass was detected which arose from the posterosuperior wall of the external auditory canal under the skin and clogged the canal .The mass was consistently soft, without any ulcer, discoloring, or bleeding on touching. The probing demonstrated no connection between mass and tympanic membrane; moreover, the eardrums were intact. The vascular appearance of the lesion was suggestive of the provisional diagnosis of granulation tissue and vascular tumor. Therefore, after examination, the radiological investigation was requested and Computed Tomography (CT) scan demonstrated a smooth mass outside the tympanic membrane ,3. No evidence of bony erosion was noted, the middle ear cavity and the ossicles looked normal, and no middle ear lesions were observed. The pre-operative audiogram illustrated unilateral moderate conductive hearing loss.The patient with an initial diagnosis of vascular tumor underwent general anesthesia and the transcanal surgical excision at the specialist discretion. The mass was dissected from the skin and sent to the pathologist, no bony erosion was noted intraoperatively, and the skin was returned back properly with no defect. The tympanic membrane was kept intact. According to the pathological investigation results, the mass was a capillary hemangioma in the external auditory canal.Finally, the patient was discharged in good general health after a successful recovery without any side effects. Postoperatively, the site of surgery healed well and the conductive hearing loss was disappeared. The postoperative audiogram recorded normal hearing ability one month after the surgery. Furthermore, no significant mass was observed on post-operative otoendoscopic examination. In addition, a one-year clinical follow-up demonstrated no recurrence of the lesion and symptoms.The external auditory canal is a segment of the external ear consisting of two parts of inner and outer. The inner ear is bony and surrounded by temporal, tympanic, and mastoid bone, while the outer ear is cartilaginous, leads to the auricle, and has thick skin with hair follicles, sebaceous glands, and ceruminous glands. The lesions of the external auditory canal can be classified as benign and malignant lesions according to their severity. Benign lesions have a higher prevalence than malignant ones .HemangioTherefore, hemangioma usually forms at birth, mostly passes the regressive phase, and disappears within the first year of life. Nonetheless, those cases which do not complete the second phase may become symptomatic and should undergo invasive investigation . Histologically, there are different types of hemangiomas, including cavernous, capillary, and mixed types. Hemangiomas are relatively common in the head and neck; nonetheless, they are very occasionally observed at the external ear canal and tympanic membrane . The revThe etiology of hemangioma in the ear canal is not well recognized; however, previously conducted studies claimed that some predisposing factors, such as trauma, infection, gender, and hormonal factors, can affect its formation and development ,8. The hApart from radiologic investigations, the temporal bone CT scan is the first choice to assess these types of tumors, providing useful information on the presence of any bony erosion. Hemangioma has the same density as brain parenchyma in CT scan results; moreover, magnetic resonance imaging (MRI) can be performed to precisely differentiate the vascular lesions. Hemangiomas present as intermediate signal T1 and hyper signal T2 in MRI. The angiography with embolization is recommended for advanced lesions .Differential diagnosis of hemangioma of the external auditory canal includes squamous cell carcinoma, basal cell carcinoma, spindle cell carcinoma, amelanotic melanoma, true hemangioma, and common warts. The treatment is complete surgical excision through a trans-canal, end-aural, or posterior auricular approach. Nevertheless, embolization or radiation therapy may be recommended in special situations . RecurreAlthough external auditory canal hemangiomas are rare, it is important to consider them in the differential diagnosis of external auditory canal masses.  Contrast-enhanced CT or MRI with gadolinium helps to reveal the vascular nature of the mass. The biopsy of vascular tumors, such as hemangioma, is not recommended since it may cause excessive bleeding. Complete surgical excision is the best choice of treatment with a minor risk of hearing impairment.


Neoplasm in South American camelids (SAC) are commonly described. The most frequently reported type of neoplasm are lymphomas and difference in the age suffering from lymphomas of and llamas is seen.This report describes a case of a solitary lymphoma in a 5 years and 9 month old llama mare displaying the approach of diagnostic imaging and successful surgical treatment.The llama was referred to the clinic for dyspnoea and inspiratory abnormal respiratory sounds. The clinical examination comprised blood cell count, ultrasonographic and radiographic examinations, endoscopy and fine needle aspiration cytology of a mass detected in the mid cervical region. The mass was surgically removed. Histopathological examination of the surgically removed mass diagnosed a malignant T-cell- lymphoma. According to the results of the clinical, ultrasonographic and radiographic examinations no tumor invasion was apparent in distant organs and the llama was discharged from the clinic seven days after surgery.Lymphoma has been reported to be the most common neoplasia in camelids and are more often described in young alpacas and in adult llamas. To the author´s knowledge the case presented here is the first that described a broad panel of diagnostic tools including ultrasound, radiographs, endoscopy, fine needle aspiration cytology and histopathoogical examination as well as a successful surgical treatment of a solitary lymphoma in camelids. In South American camelids (SAC) neoplasms are commonly seen –6. The mA 5 year and 9 month old, 170 kg weighing (body condition score (BCS) 3.5) domestic llama mare was referred to the University Clinic for Ruminants at the University of Veterinary Medicine Vienna due to dyspnoea and inspiratory noise for one week. An esophageal obstruction had been suspected by the referring veterinarian, however administration of spasmolytics and passing of a tube had not improve the symptoms.The mare was bright, alert, in good general condition and had a BCS of 3.5 out of 5. In the middle third of the ventral cervical region, a protruding mass was present. The mass was 10 × 8x3 cm, partially firm and fluctuant on palpation, slightly painful and the palpable local temperature was mildly increased. The overlying skin appeared normal. No further masses or external lymph node enlargement was detectable. There were no signs of dysphagia. Besides these findings all other vital parameters were within normal ranges. Blood cell count showed mild leukocytosis, neutrophilia, lymphopenia and eosinopenia. Haematocrit  and MCV  were mildly increased, MCHC  was mildly decreased. TWBC was increased . Segmented neutrophils were mildly increased , Lymphocytes  and Eosinophils  were moderate decreased. All other values were within normal limits.Flexible endoscopy  was performed in the sedated llama , butorphanol , each 0.2 mg/kg, IM) via ventral nasal meatus in order to view the esophagus. No pathological findings were detected over the entire length of the esophagus, the mucosa was whitish and smooth, peristaltic movements were seen. Endoscopic examination of the trachea and airways was inconspicuous.Ultrasonography was performed with a 14 MHz linear probe used in trapezoid fashion (Samsung HS60). The mass presented subcutaneously and well demarcated from the surrounding tissue with heterogenic echogenicity and a firm capsule Fig. . A neoplLatero-lateral and dorso 35°left lateral- ventrolateral oblique radiographs of the mid to caudal cervical third were performed by the use of a CR System FCR IP Typ CC Pb 24 × 30 cm  and a mobile radiographic machine . A ventrally well and dorsally ill defined, homogenic mass of soft tissue density could be depicted. A mild mass phenomenon to the left ventral aspect of the trachea was visible. All other structures were within normal ranges. Latero-lateral radiographs  of the thorax showed no pathological signs related to the mass , for the skin a combination of vertical U and simple interrupted sutures with polypropylene No. 0 and 1 was used.A stent bandage was sutured over the surgical site and covered with adhesive bandage material. The incision healed uneventfully and the sutures were removed 12 days after surgery.Histopathological examination of the surgically removed mass was performed. The size of the fixed tumor specimen excised from the middle of the ventral, cervical region was 7 x *6 x *2.5 cm and of firm and elastic consistency. The inhomogeneous cut surface consisted of a beige to yellowish soft tissue with oligofocal, clearly demarcated, irregular formed areas of whitish colour. After fixation for 24 h in 4% neutral buffered formaldehyde, the sample was dehydrated in rising concentrations of ethanol and embedded in paraffin wax. Sections of 4 µm thickness were stained with hematoxylin and eosin (HE) (Figs. Histopathological investigations confirmed a highly cellular, nodular tumor mass consisting of lymphoid cells accompanied by sparse amounts of fibrovascular stroma Fig. , thorougImmunohistochemical analysis displayed that the neoplasia was composed of diffuse, numerous CD3 immunolabelled cells Fig. b and intNeoplasia are commonly diagnosed in SAC. As their popularity is still ongoing and mostly kept as pets in many countries, they often reach a higher age than other ruminants , 13. ThiThis is consistent with the age of the patient described, who was 5 years and 9 months old at the time of diagnosis.Considering age, malignant round cell neoplasms can be differentiated by their immunohistological profiles and locations. T cell lymphoma in SAC have been reported albeit less frequently than B cell lymphomas . LymphomIn a study investigated 110 camelid neoplasm and diagnosed lymphoma and/or leukemia in 20 alpacas and 6 llamas . In the Common clinical signs are often nonspecific and highly variable like tissue enlargement in different palpable areas, sizes or shapes, anorexia, weight loss, poor growth, weakness or recumbency , 8. In tDiagnostic workup associated with neoplasms has been widely described in case reports or case series , 8. DuriDuring clinical examination and diagnostic imaging no metastasis or lymphadenomegaly could be found, although lymph node enlargement is a common symptom , 10, morThis study and previous studies indicate that camelids with MRCT can present with a variety of clinical signs and laboratory findings, and that the clinical course is variable. This case report points out, that an early detection followed by surgical procedures can expand the life span of the affected animal.