Gastrointestinal bezoars may occur in individuals with a normal gastrointestinal tract structure or as a result of gastrointestinal defects and disease. This rare condition initially presents with general abdominal pain, mimicking appendicitis in later stages. Recognizing this condition as a differential diagnosis in patients with abdominal pain can prevent delays in diagnosis and serious complications.We report a rare case of a meat bezoar in a 52-year-old man presenting with acute and generalized abdominal pain at an emergency department.We discuss gastrointestinal bezoars as a rare differential diagnosis of abdominal pain and acute abdomen in people with no pre-existing medical history, and the challenges that might be faced during diagnosis and treatment.Gastrointestinal bezoars are rare which makes the diagnosis challenging. Obtaining a complete history and a full examination with appropriate imaging could help the diagnosis. •Bezoars are rare and usually show no symptoms that make them underdiagnosed in cases with abdominal pain.•Bezoars can be symptomatic and cause serious complications with delay in diagnosis.•Symptomatic bezoars may be misdiagnosed with appendicitis.•Obtaining an accurate history of patients with abdominal pain is one of the most important parts of diagnosis for bezoars.•Imaging will help diagnose bezoars in patients with general symptoms. While t2A 52-year-old man with no significant past medical history presented to the emergency department of Firuzabadi Educational Hospital in Tehran, Iran, with acute, generalized abdominal pain. The patient was not taking any medications, and there was no medical history suggesting an underlying condition related to chewing disorders. The patient reported no nausea, vomiting, or other gastrointestinal symptoms except for obstipation over the past three days. There was no tenderness, guarding, or rebound tenderness in the initial examination.Laboratory assessment yielded normal results without any evidence of leukocytosis, raised inflammatory biomarkers, or abnormalities in urinary analysis. Standing abdominal and pelvic radiographs were normal showing no signs of pneumoperitoneum or air-fluid levels. Abdominal and pelvic ultrasound showed an inter-loop fluid collection in the right lower quadrant. Accordingly, the patient was admitted to the hospital.During hospitalization, a serial examination of the abdomen was performed in terms of the presence of tenderness, rebound tenderness and guarding, all of which were normal. Serial rectal examination also revealed no abnormalities. However, after 24 h of hospitalization, the patient's abdominal pain intensified and localized to the right lower quadrant. Physical examination now revealed mild abdominal distension and right lower quadrant tenderness and rebound tenderness. Additionally, the patient had not tolerated oral intake for the past 12 h and had not defecated during hospitalization.Repeated abdomino-pelvic ultrasound showed dilated jejunum loops with no evidence of appendicitis. In our center, CT-san capabilities were unavailable. Due to severe abdominal pain and tenderness in the right lower quadrant, the patient's obstipation and oral intake intolerance, with suspicion of intestinal obstruction, the patient was immediately transferred to the surgery room and underwent exploratory laparotomy through a midline incision. During exploratory laparotomy, dilated small intestine loops with intraluminal masses in the ileum terminal were discovered. The ileum diameter before and after the obstruction site measured 5 cm and 1 cm, respectively. Additionally, necrosis, and sealed perforation were found 50 cm proximal to the ileo-ceal valve, where an Enterotomy was performed. The Enterotomy revealed large bezoar masses in front of the ileocecal valve, causing partial bowel obstruction . A samplPostoperative recovery proceeded smoothly. The patient was initially transferred to the intensive care unit and, on the second post-surgery day, to the surgery ward in stable condition. Due to mild ileus in the early post-operative period, a normal diet was not initiated until the fifth day after the surgery. On the fifth post-surgery day, the patient successfully tolerated a normal diet, and the abdominal skin was closed on the sixth post-surgery day. The patient was discharged the following day. The patient's medical history revealed that he had ingested Chenjeh Kebab, a kind of Persian roasted lamb pieces. According to the pathology report, inadequately masticated met pieces were identified as the etiology of bezoar formation. The patient underwent a follow-up two months later, and no clinical problems were observed.The patient provided informed consent for reporting his condition and treatment approaches for educational and academic purposes.3While very rare, cases of meat bezoars have been previously described. In a case presented by Henry et al. in 2016 , a 53-yeIn our study, the patient initially presented with generalized abdominal pain. Despite normal laboratory and imaging reports, ultimately, the bezoar was diagnosed through exploratory laparotomy. Bezoars can consist of different components, such as indigestible vegetable or fruit fibers, hair, food particles, milk proteins, and even medications, resulting in varied presentations and side effects. To effectively manage gastrointestinal bezoars, especially, those related to inadequate food mastication, obtaining a detailed and accurate medical history is crucial. Bezoars may be asymptomatic or present with symptoms such as a palpable abdominal mass, gastric outlet obstruction, gastric pain, early satiety, weight loss, and even ulceration, perforation and hemorrhage. In some cases, the symptoms may be vogue or nonspecific, leading to a misdiagnosis or delayed diagnosis . Since mAbdominal radiography has been found useful in suspecting bezoars in approximately half of the affected patients . UltrasoThe meat bezoar in this case formed due to inadequate mastication without any pre-existing medical conditions. The delayed definitive diagnosis could have led to adverse outcomes. Nevertheless, the patient's clinical condition improved after the surgery, and he remained asymptomatic thereafter.4This study has certain limitations that need to be acknowledged. Firstly, it is based on only one case, making it difficult to generalize the symptoms and findings to all other cases with similar presentations. Therefore, the findings should be interpreted with caution and considered more as a guiding case in its specific context.Additionally, the study lacked an explicit scientific method for case presentation. To address this limitation, we followed the Updating Consensus Surgical CAse REport (SCARE) 2018 guideline, ensuring rigor in reporting and analysis .5Despite being a rare diagnosis in patients with abdominal pain, gastrointestinal bezoars should always be considered as a possible differential diagnoses due to their potential to cause bowel obstruction and serious complications. To effectively manage such cases, obtaining a complete medical history is essential, as it can guide the selection of appropriate approaches and diagnostic tests for these patients.Ethical approval is not required for this study design in our institution.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Tahmineh Tahouri: Study design and concept, supervising accuracy of data collection, data validation, and data analysis.Ehsanollah Rahimi-Movaghar: Study design and concept, data collection and following-up, data analysis and interpretation, manuscript accuracy checking.Mohammadreza Safaei Qomi: Data collection, writing and editing the manuscript.Sarvenaz Mehrabi: Literature review, writing and editing the manuscript, manuscript accuracy checking. All authors confirmed the final manuscript and accepted its publication.Dr. Ehsanollah Rahimi-MovagharThe patient was informed about the intention to report his condition and the treatment approaches for educational and academic aims, and written informed consent was obtained for the publication of this case report.The authors declared no conflict of interest.


Subglottic hemangiomas are rare in adulthood. The presence of the lesion in the subglottic region makes it even more unusual. Moreover, these lesions do not have a typical course and involution changes as seen in the infantile forms. An elderly female initially came with a brief history of dyspnea and symptoms of upper respiratory tract infection. The patient also complained of a change of voice and noisy breathing, with a recent history of intubation following COVID-19 pneumonia and late-onset bronchial asthma. Flexible nasopharyngolaryngoscopy showed a mass below the vocal folds, which was seen to arise from the posterior subglottic region. The patient eventually underwent endoscopic excision of the lesion under general anesthesia and recovered well. Symptoms of hoarseness and stridor, along with a history of intubation, should raise a high index of suspicion for laryngeal diseases. A delay in the diagnosis of an obstructing lesion in the subglottis occurs in the presence of a confounding lung infection and overlap of clinical features with those of bronchial asthma. Surgical excision is required not only to alleviate obstructive symptoms but also to rule out malignancy. Hemangioma is the most common congenital benign tumor of the larynx, which is rare in adults. It is a vascular malformation that is slow-growing and self-limiting in nature. Hemangiomas can be classified either by the age of occurrence (there are infantile and adult forms), their anatomical location, or their histological characteristics. In their infantile form, these lesions demonstrate a characteristic clinical course marked by rapid proliferation, slow and spontaneous involution, and then resolution . As suchMeanwhile, in adults, hemangiomas act rather differently and do not follow the usual phases that occur in children. The lesions are usually non-progressive. In adults, the most frequent location for laryngeal hemangiomas is the supraglottic region (80%), followed by the glottic and subglottic regions . These vHereby, we would like to share our experience in dealing with a very rare case of subglottic hemangioma presenting in an elderly female with a co-existing lung infection and bronchial asthma.A 73-year-old lady with multiple comorbidities presented to the casualty with a brief history of difficulty breathing for a day. She also had symptoms of an upper respiratory tract infection. The patient also complained of a change of voice for the past month. The voice changes developed post-extubation, where she was electively intubated for five days and kept sedated in the intensive care unit due to coronavirus disease 2019  pneumonia. There was no history of change of voice or hemoptysis prior to that. She was admitted to the medical ward and treated for community-acquired pneumonia with concomitant newly diagnosed bronchial asthma. The patient was then referred to the otorhinolaryngology team on the second day of admission for upper airway assessment. Upon assessment, there was soft biphasic stridor with no signs of respiratory distress, and a flexible nasopharyngolaryngoscopy (FNPLS) showed a reddish mass below the vocal folds, which was seen to arise from the posterior subglottic region. The supraglottic structures and vocal folds were normal. In view of a recent history of intubation, the initial diagnosis of acquired subglottic stenosis was made. The patient was started on nebulized dexamethasone and ciprofloxacin 12 hourly, intravenous dexamethasone eight hourly, and intravenous cefepime to treat underlying pneumonia. In view of the non-resolving symptoms, the patient underwent computed tomography (CT) of the neck, which reported a well-defined hypodense submucosal lesion with peripheral enhancement at the left posterior paramidline subglottic region, measuring approximately 0.7 x 1.1 x 1.0 cm. The adjacent cricoid cartilage was eroded with no extralaryngeal extension or suggestion of lymphatic metastasis Figure . The patAdult subglottic hemangiomas are scarcely reported in the literature. The common attributable factors for laryngeal hemangiomas are cigarette smoking, vocal abuse, and laryngeal trauma such as intubation . In thisAnother important differential diagnosis that ought to be considered in any adult with stridor or upper airway obstruction is laryngeal carcinoma, especially in the elderly group. Thus, surgical intervention is necessary not only to alleviate the symptoms suffered but also to attain a histopathological diagnosis to distinguish the lesion from any malignancy. In laryngeal carcinoma, the main symptom is also hoarseness, as experienced by the patient discussed in this case report. Other significant symptoms include a persistent sore throat, dysphagia, and dyspnea. The duration of each of the symptoms can also differ. According to a retrospective study done in Finland in 1999, hoarseness generally has the longest duration of any symptom, whereas more worrying symptoms like dyspnea and hemoptysis have the shortest durations .The management of laryngeal hemangiomas in adults is mostly surgical, as these vascular lesions do not regress spontaneously. Generally, smaller hemangiomas can be treated with laser ablation, corticosteroid injections, and cryosurgery. On the other hand, large lesions with extension to deeper structures require endoscopic laryngeal surgical excision or radiotherapy ,8. In thAdult subglottic hemangioma is a very rare entity that can occur in patients with hoarseness and respiratory distress. Unresolved co-existing lung infections with a history of intubation should raise a high index of suspicion for these types of rare lesions. Surgical excision is required not only to alleviate obstructive symptoms but also to rule out malignant diseases in the larynx.


The study reports a rare case of pelvic solitary fibrous tumor (SFT) that was initially considered as a peri‐anal gastrointestinal stromal tumor due to similar radiologic/pathologic features. SFT diagnosis can be challenging due to its rarity and wide range of diagnoses that must be ruled out precisely.Solitary fibrous tumors (SFTs) are rare tumors that can occur in any part of the body. Although usually benign, malignant SFTs have been reported, especially outside the lungs. Radiology can help with diagnosis, but immunohistochemistry is necessary to distinguish SFTs from other possible diagnoses such as gastrointestinal stromal tumors (GISTs). This study presents a rare case of pelvic SFT initially considered to be a peri‐anal GIST, highlighting the importance of accurate diagnosis given the rarity of SFTs and the need to rule out other potential diagnoses. Resected pelvic solitary fibrous tumor (SFT). The patient had no significant symptoms except slight generalized abdominal pain and peri‐anal discomfort and she did not state any previous experience of similar abdominal or pelvic pain and GI disorder in herself or her family members. All of the examinations including abdominal and rectal examinations were normal without any suspicious findings.All of the patient's medical data regarding CT scan, magnetic resonance imaging (MRI), sonographies, and angiography prior to angioembolization in addition to the final pathology and IHC results of the resected tumor, are provided in order in the following parts.First, a spiral thoraco‐abdomino‐pelvic CT scan with and without contrast was performed in which an infiltrative mass with the central necrotic components was found in the left peri‐anal region pressing the external anal sphincter. The mass had the size of about 96 × 66 × 48 mm growing to the perineal area causing superior displacement of levator ani on the left side; still, it had not shown any developments to the peri‐rectal region and is limited to the perineal region. Blood supply of the mass was provided by the left pudendal artery. No invasion to the muscles, bones, and other peripheral structures was mentioned and all other organs and structures were completely intact Figure . After tAfter complete tumor resection Figure , the pel3SFT is a spindle‐cell fibroblastic mesenchymal neoplasm that can affect almost all anatomic sites but most commonly occurs in visceral pleura.In addition to the rarity of this neoplasm, SFT has a wide range of differential diagnoses that make accurate SFT diagnosis more challenging. Indeed, imaging studies along with considering related clinical features may lead to SFT diagnosis; but a definite diagnosis should be confirmed through IHC evaluations in order to differentiate it from other mesenchymal tumors with similar sources.Complete resection is the choice treatment strategy for SFTs that is connected to patients' prognosis4SFT can be introduced as a rare and slow‐growing metastasizing fibroblastic mesenchymal neoplasm having a variety of differential diagnoses and can mimic other tumors' characteristics. Although its diagnostic processes may benefit from radiologic evaluations in the first steps, IHC studies have shown great importance in distinguishing it from other diagnoses due to diagnostic challenges.Seyed Amir Miratashi Yazdi: Conceptualization; writing – review and editing. Roudsari Payvand Parhizkar: Conceptualization; writing – original draft; writing – review and editing. Hadi Ahmadi Amoli: Conceptualization; supervision. Reza Hajebi: Conceptualization; writing – review and editing.This study received no funding or financial support.The authors declare that there is no conflict of interest.The study was performed regarding the ethics committee of Tehran University of Medical Sciences.The authors have confirmed that patient consent has been signed and collected in accordance with the journal's patient consent policy.


Anisakis sp. larvae and is extremely rare, especially when originating in the esophagus. We present a case of esophageal anisakiasis in a 61-year-old male who experienced severe precordial pain and radiating discomfort to the neck after consuming raw fish sashimi. Upper gastrointestinal endoscopy revealed the presence of a larva in the upper esophagus. On the basis of anatomo-morphological features, the worm was provisionally identified as Anisakis sp. and was easily extracted with forceps, which led to a prompt improvement in the patient’s symptoms. This case highlights the importance of considering anisakiasis as a differential diagnosis in patients with gastrointestinal symptoms and a history of consuming raw fish.Anisakiasis is caused by consuming raw fish contaminated with  Anisakis sp. larvae. Patients’ symptoms range from mild abdominal pain to severe complications, such as a bowel obstruction or perforation. Reports of this disease are frequent in Japan, where the consumption of raw seafood is common. However, most cases originate in the stomach, while esophageal anisakiasis is extremely rare [Anisakis sp. larva in the upper esophagus of a patient.Anisakiasis is a parasitic gastrointestinal disease that causes various gastrointestinal symptoms and can occur after the consumption of raw or undercooked seafood infected with ely rare . We descLateolabrax japonicus, Salmo salar, Seriola quinqueradiata, and Lutjanus stellatus). The patient experienced a gradual increase in radiating pain to both sides of the neck, prompting him to visit the emergency department of our hospital the next day. The patient had no significant medical history or known allergies. He was 175 cm tall and had a healthy weight of 65 kg. A physical examination revealed a body temperature of 36.6 °C, blood pressure of 146/87 mmHg, a respiratory rate of 12 breaths per minute, an SpO₂ value (room air) of 99%, and was alert. Upon the auscultation of the chest, breathing sounds were found to be normal, no significant heart murmurs were detected, and no spontaneous or palpable pain was observed in the upper abdomen. An electrocardiogram did not reveal any signs of ischemic heart disease. We subsequently suspected anisakiasis based on the patient’s history of consuming raw fish and performed an esophagogastroduodenoscopy (EGD) using a GIF-H290 videogastroscope  on the same day. The EGD revealed the presence of a worm-like structure, approximately 20 mm in length, embedded in the mucosa of the upper esophagus, approximately 25 cm from the incisors (Anisakis sp. larva by two gastroenterologists at Juntendo University (Eiji Kamba and Takashi Murakami). The molecular identification of the nematode was not performed. Acute mucosal lesions, other than the Anisakis sp. larva, or vanishing tumors were not found in the rest of the esophagus, stomach, and duodenum. The Anisakis sp. larva was swiftly and easily removed using biopsy forceps  in which they develop into third-stage larvae. These can be passed on to fish and squid, and become infectious to humans when accidentally ingested . SalmoniCurrently, nine known species exist within the Anisakis sp. genus, and their third-stage larvae are classified into four types, namely, Types I to IV, based on morphological differences . Of thesWhile gastrointestinal anisakiasis has been extensively reported in Japan, where a tradition of consuming raw seafood exists, the consumption of raw fish has increased in Western nations and caused a rise in the incidence of clinical cases of anisakiasis ,13. The Anisakiasis can be categorized into three distinct types: gastric, intestinal, and ectopic . The mosThe clinical manifestations of anisakiasis are not only confined to gastrointestinal symptoms, such as nausea, vomiting, and epigastric pain, but also include allergic reactions to the penetration of Anisakis sp. larvae into the gastrointestinal wall, including angioedema and urticaria. It is common for the condition to be accompanied by the presence of ascites or symptoms indicative of peritoneal irritation. In cases of small intestinal anisakiasis, the condition can also lead to symptoms of intestinal obstruction or intestinal intussusception . From a In the context of gastric anisakiasis, diagnosing the condition using endoscopic imaging is relatively straightforward since whitish, thread-like, worm-like bodies can be observed. Apart from local redness, edema, and ulceration, the presence of such worm-like bodies can cause inflammation in the submucosal layer, resulting in a tumor-like elevation. This elevation, often referred to as a “vanishing tumor”, tends to resolve within approximately 1 to 2 weeks along with an improvement in the inflammation. Approximately 2–4% of gastric anisakiasis cases present with a vanishing tumor . In someRegarding the mechanism of Anisakis sp. larvae penetrating the esophagus, Urita et al. postulated that larvae initially entered the stomach and then returned to the esophagus by gastroesophageal reflux, leading to esophageal anisakiasis . HoweverThe mainstay of treatment is an early endoscopic extraction. It is important to grab a larva as close to its embedded portion as possible to ensure no part of the larva remains within the mucosa. If not removed completely, the larva can cause chronic inflammation leading to various gastrointestinal symptoms. Conservative treatment with anti-allergic medication is considered effective against anisakiasis. However, it should be noted that the presence of invading larvae can lead to granuloma formation . AdditioAlthough esophageal anisakiasis is an extremely rare condition, it should be considered during the differential diagnosis of patients presenting with gastrointestinal symptoms and a history of consuming raw fish. Obtaining a detailed food history will often be the key to a diagnosis as symptoms usually arise shortly after the ingestion of food contaminated with the parasite.


Infiltrative lipomas have a tendency to infiltrate adjacent muscle and fibrous tissue without metastasis, such as muscle; connective tissue; bone; and, in rare cases, peripheral nerves and the spinal cord. Incomplete surgical excision of infiltrative lipoma has been shown to increase recurrence. This case report describes a dog with an extensive infiltrative lipoma impinging on the vertebral canal, compressing the spinal cord and extending into the surrounding muscle layers and thoracic cavity. The mass was surgically removed, and subsequent postoperative computed tomography with the automated fat measurement technique confirmed complete removal. The patient’s prognosis could be assessed through postoperative computed tomography, and there has been no recurrence during the 2-year follow-up period.® advanced visualization fat measurement. Histopathological analysis confirmed that the mass was an infiltrative lipoma. The patient’s symptoms completely resolved after surgery, with no recurrence reported at the 2-year follow-up. This case highlights the benefits of using postoperative computed tomography combined with the automated fat measurement technique to determine whether reoperation is necessary or to predict patient prognosis by identifying potential residual lipoma post-surgery.Infiltrative lipomas, which are locally invasive tumors composed of well-differentiated adipocytes, are histologically identical to lipomas but have a tendency to infiltrate adjacent muscle and fibrous tissue without metastasis, such as muscle; connective tissue; bone; and, in rare cases, peripheral nerves and the spinal cord. They differ from liposarcomas yet also exhibit neoplastic cell infiltration and often recur despite surgical removal. A 10-year-old spayed Maltese female dog presented with hindlimb paresis and back pain for two months. Computed tomography and magnetic resonance imaging revealed an extensive fatty mass impinging on the vertebral canal, compressing the spinal cord, and extending into the surrounding muscle layers and thoracic cavity. The mass was surgically removed, and subsequent postoperative computed tomography confirmed complete removal of the mass using Vitrea Infiltrative lipomas, which are locally invasive tumors composed of well-differentiated adipocytes, are histologically identical to lipomas but have a tendency to infiltrate adjacent muscle and fibrous tissue without metastasis, such as muscle; connective tissue; bone; and, in rare cases, peripheral nerves and the spinal cord ,2,3. It Advanced imaging techniques are essential for accurately assessing the distribution, shape, size, and location of the mass before surgical intervention or radiation therapy . ComputeThe present case report describes the removal of an extensive infiltrative lipoma within the thoracic vertebral canal, whereby complete excision was confirmed using CT immediately after surgery.™; GE Healthcare, Seoul, Republic of Korea) was administered into the cephalic vein at a rate of 2.0 mL/s via a power injector . The CT images revealed a well-defined, homogeneous, hypoattenuating mass with a density of −119 ± 8 Hounsfield units (HU), indicative of lipoma. This fatty mass also exhibited extensive distribution spanning from T3 to T5, measuring approximately 2.3 cm (height) × 1.9 cm (length) × 1.7 cm (width), and in the vertebral canal and surrounding muscle layers on the left side of T4. Moreover, destruction of the left T3–T5 pedicles and T3 and T4 vertebral bodies was observed, with subsequent invasion into the thoracic cavity , raising suspicion of an aggressive lesion, such as a tumor . Pre- anc cavity . Three-dMRI was performed using a 1.5 T MRI scanner  with 3 mm slice thickness to obtain detailed information about the spinal cord and soft tissues surrounding the fatty mass in the thoracic vertebrae. T1- and T2-weighted MRI revealed a hyperintense mass that extended into several muscle layers surrounding the left side of T4  . On post® Advanced CT fat measurement) for confirming the complete removal of adipose tissue immediately following surgical excision.This report describes a case of an infiltrative lipoma with extensive neurological symptoms involving the vertebral column and surrounding muscle tissues and spinal cord compression and demonstrates the clinical usefulness of postoperative CT with the automated body fat measurement technique (VitreaOn the CT images, the fatty mass in this case exhibited a uniform hypoattenuating fat density without contrast enhancement and had infiltrated the surrounding muscle layers extensively, leading to a strong suspicion of an infiltrative lipoma rather than a lymphosarcoma or a simple lipoma. In this case, the infiltrative lipoma led to thoracic vertebra destruction and spinal cord compression. In the literature, seven cases of infiltrative lipoma invading the vertebral canal and causing neurological deficits have been documented ,5,6,7,10Although MRI is more sensitive than CT for detecting microscopic fat, the scanning time is significantly longer; hence, postoperative CT was performed to avoid extending the anesthetic duration. Nonetheless, CT imaging with automated body fat measurement helps distinguish fat masses with greater sensitivity compared to standard CT methods, highlighting the clinical utility of this technique and its potential as a viable alternative to MRI.While the implementation of intraoperative CT scanning has been reported in human medicine , researcNevertheless, this study has some limitations, which include the potential for recurrence in the future, and follow-up CT scans after discharge have not been conducted thus far. Despite the patient undergoing complete lipoma resection and remaining free of recurrence for 2 years, whether vertebral body lysis was a direct consequence of bone infiltration or secondary to pressure-related mechanisms, such as vascular supply compromise, remains unclear, limiting prognostic accuracy . HistopaThe use of postoperative CT with automatic fat measurement facilitated the complete removal of an infiltrative lipoma extending into multiple muscle layers and the vertebral canal in a dog. This procedure is particularly advantageous over conventional CT during the removal of masses with extensive infiltration or those located in challenging areas that cannot be visualized during surgery. It can also help predict patient prognosis by identifying potential postoperative residual lipomas and determining whether reoperation is necessary. While the approach of combining automatic fat measurement was highly beneficial in this case, it is imperative to assess and determine its clinical utility by applying it to a larger cohort of veterinary patients.


Amyloidosis is the process of extracellular deposition of protein fibrils and manifests pathologically as a systemic or localized process. Localized amyloidosis of the head and neck is uncommon, and involvement of the sphenoid sinus is exceedingly rare. We describe a case of localized amyloidosis isolated from the sphenoid sinus. A descriptive literature search was conducted to highlight presentation, management, and outcomes related to this pathology. Our patient was a 65-year-old male who presented to our clinic with nasal congestion and an incidental finding of a large expansile mass within the sphenoid sinuses. The mass was seen to displace the pituitary gland, and thus a multidisciplinary care approach ensued. The mass was removed via a transnasal endoscopic approach. Pathology revealed fibrocollagenous tissue with calcifications that were positive on Congo red staining. The patient underwent further workup to rule out systemic involvement, which was unremarkable. Based on the findings of his workup, he was ultimately diagnosed with localized amyloidosis. A comprehensive review of the literature revealed 25 other reported cases of localized amyloidosis within the sinonasal region, with only one other case of isolated sphenoid sinus disease. Common presenting symptoms are nonspecific and may mimic other, more frequently seen regional pathologies, including nasal obstruction, rhinorrhea, and epistaxis. The treatment for localized disease is surgical resection. While localized amyloidosis within the sinonasal region is rare, it is important to recognize, work up, and treat it appropriately. A multidisciplinary team approach is necessary for appropriate diagnosis and management, and these patients should be followed closely after treatment. Amyloidosis refers to the extracellular tissue deposition of misfolded protein fibrils. These deposits may result in a variety of clinical manifestations, depending primarily on the type, location, and quantity of protein aggregates ,2. SeverClinicoradiographic presentation A 65-year-old male was referred to the otolaryngology clinic after incidental findings on imaging. He initially presented to his primary care provider with nonspecific symptoms, including nasal congestion, mild left facial pressure, and sleep disturbances. His medical history was significant for hypertension, hypercholesterolemia, and continuous positive airway pressure (CPAP)-compliant obstructive sleep apnea. He had an elevated body mass index (BMI) of 37 and was a nonsmoker with a moderate daily alcohol intake. Physical examination, including nasal endoscopy, revealed a severely deviated septum to the left and bilateral inferior turbinate hypertrophy but was otherwise normal. A computed tomography (CT) scan without contrast of the sinuses was ordered by the referring provider, incidentally showing a large expansile intermediate density lesion centered in the sphenoid sinuses with extensive bony remodeling Figure . MagnetiSurgery Given the patient's clinical and radiographic findings, the decision was made to proceed with surgery. At this point, the differential diagnosis included an expansive mucocele, sphenoid allergic fungal sinusitis, and a pituitary mass, with the former being the most favored. The patient was taken to the operating room for a septoplasty and endoscopic sinus surgery, with a focus on debulking the mass. The sphenoid sinus os was identified and widened, revealing a large soft tissue mass with a fibrous capsule filling the entire sphenoid sinus. There was no intersphenoid septum, and the right sphenoid sinus appeared hypoplastic. The mass was removed entirely using a Kerrison rongeur, apart from a small cuff that was left in place over a dehiscent lateral wall. Thick mucous secretions and calcified fragments were also encountered and removed from within the fibrous capsule. These findings initially raised suspicions about fungal debris. A frozen section intra-operatively showed fibrous tissue with scattered chronic inflammation, scant mucinous material, and respiratory-type epithelium.Due to the displacement of the pituitary gland and pituitary stalk, an endoscopic evaluation of the pituitary region was performed by the neurosurgeon. The floor of the sella was noted to be eroded by the mass; however, the area appeared to be adequately decompressed and composed of a normal-appearing pituitary gland, which was preserved.Post-operative course The final pathologic evaluation of the sphenoid mass showed fibrocollagenous tissue with crushed cells and dystrophic calcifications. A Congo red stain of the biopsy was positive and therefore determined to be involved in amyloidosis of indeterminate protein type Figure . ProlactThe patient was seen at four months and one year postoperatively with significant improvement in his nasal congestion and overall breathing. There were no interval complaints or complications noted. The patient’s prolactin levels ultimately improved, and the cabergoline dose was decreased, which is managed by the Endocrinology team. The testosterone levels also improved postoperatively, and the patient was not started on other long-term medications. He will continue to follow up with his multidisciplinary team. Localized amyloidosis of the head and neck is a rare entity and most frequently involves the larynx, pharynx, thyroid gland, and trachea -6. The sLocalized amyloidosis within the sinonasal region more frequently affects women than men and affects all age groups . PatientThe diagnosis of amyloidosis is determined via Congo red staining of the tissue biopsy. Further classification as systemic or localized amyloidosis is critical, as systemic amyloidosis is associated with significant morbidity and mortality, and its management involves the administration of chemotherapeutic and anti-inflammatory agents. In contrast, localized amyloidosis can be effectively managed surgically with an excellent prognosis . To deteLocalized disease is typically treated with complete surgical resection . In highLocalized amyloidosis in the sinonasal tract is exceedingly rare and may manifest with non-specific symptoms commonly shared with other pathologies in this region. Early assessment and detection are necessary for the proper management of these patients, and a multi-disciplinary approach is imperative in ruling out systemic disease. We shared the case of our patient with localized sphenoid sinus amyloidosis, who was managed successfully with surgery and appropriate follow-up.


Dirofilaria repensin a 4-year-old male patient in Switzerland. The disease is a vector-borne parasitic infection that is not endemic to Switzerland. A 4-year-old male presented with a tender mass in the left groin. The patient was taken to the operating room for surgical exploration to rule out a pathology that could be harmful to the spermatic cord. A node was found along the spermatic cord and excised. Histopathology and microbiology studies revealed the diagnosis ofDirofilaria repens.We report the first case ofDirofilaria repens, the diagnosis of a parasitic infection should be considered in patients presenting with subcutaneous nodules in correlation with a travel history to endemic areas. The treatment consists of complete excision of the affected tissue.Even though Switzerland is not endemic to Dirofilaria repens(D. repens) belongs to the species of filarial nematodes. Their primary hosts are dogs, foxes, and other carnivores.D. repenscannot reach the adult stage of their lifecycle in humans, they remain confined to or near the local site of infection, without the risk of a systemic infection. Humans most commonly present with subcutaneous nodes that may be accompanied by local edema, overlying redness, rash, or pain. Lesions may appear anywhere in the body and have been reported in the skin, eyes, lungs, and viscera.D. repensis mainly endemic in the Mediterranean and eastern countries of Europe, Africa, and Asia.D. repensinfection in a pediatric patient in Switzerland.A 4-year-old male patient was referred to the emergency department (ED) for a tender lump in his left groin. The lesion was present for 3 months, however, acute pain and swelling only started 24 hours prior to presentation. Upon clinical exam, the patient was afebrile with normal vital signs. An approximately 1 × 1 cm tender mass was palpated in his left groin along the spermatic cord proximal to the external inguinal ring. The overlying skin was intact, with local redness and edema. The scrotum was mildly swollen and red, with a normal clinical exam of both testicles. Initially, the ED considered the diagnosis of an inguinal hernia and attempted a reduction. It was found that the mass could not be reduced. An ultrasound of the groin demonstrated edema and hyperperfusion along the spermatic cord and epididymis, consistent with funiculitis and epididymitis. There was a small hydrocele and lack of any signs suggestive for testicular torsion. Blood work demonstrated a normal leucocyte count, normal levels of eosinophil, and increased levels of neutrophil granulocytes, along with mild lymphopenia. The patient was otherwise healthy. The family lived in a rural setting and had a dog. A travel history was positive for travelling to Croatia 4 months prior to the first presentation of the node. At that time, the patient had been stung by mosquitoes multiple times.After initial presentation in the ED, the patient was discharged home with symptomatic treatment for epididymitis. He returned within 12 hours with progressive pain and soft tissue edema. Due to progressive symptoms, the patient was taken to the operating room for surgical exploration to rule out a pathology that could be harmful to the spermatic cord. Following an inguinal incision, the subcutaneous tissue was found to be inflamed and edematous. The external oblique fascia was opened and the testis along with the spermatic cord was delivered. An encapsulated hydrocele along the spermatic cord was opened and drained amber-colored serous fluid. The testis and epididymis appeared inflamed but with otherwise normal anatomy. An indurated soft tissue nodule was found adherent to the internal spermatic fascia along the spermatic cord . The maAfter consulting infectious disease, no additional medical treatment was given. The postoperative course was uneventful.D. repens. Microbiology swaps of the hydrocele, the testis, and the lesion returned negative.The definitive histopathology of the specimen with paraffin embedding and hematoxylin and eosin staining demonstrated connective tissue with parts of a filarial nematodes . It wasD. repensalong the spermatic cord. To screen for similar reports ofD. repensin male patients, a literature review was performed using combinations ofD. repensand spermatic cord, testis, epididymis, and scrotum in PubMed. The search yielded 20 case reports, with six including children .This is the first report from Switzerland of an infection withIn the majority of cases, the pathology developed over time, commonly presenting as a solid or cystic mass. Only a few cases presented acutely, mimicking acute scrotum, testicular torsion, or incarcerated inguinal hernia. Our patient had traveled to Croatia 4 months prior to the first presentation of the node, which he had for 3 months before presenting to the ED with acute symptoms. This is consistent with previously reported symptoms, and with an incubation period of 6 to 8 months.D. repenswas made after histopathological examination of the extirpated tissue.Diagnostic blood work and ultrasound generally demonstrated similar results to our case, with oval nodules with cystic contents in selected cases. On rare occasions, radiologists were able to demonstrate linear hyperechogenic structures, sometimes with spontaneous movement, representing worms. Intraoperative localization of the pathologies was described along the spermatic cord, in and on the testis and epididymis, and in the subcutaneous tissue of the scrotum. The tissue was reported as firm nodules, cysts, or mixed solid-cystic lesions. In all cases, resection of the pathology was the treatment of choice. Knowledge of this entity is essential to the pediatric surgeon, as some surgeons performed hasty orchiectomy, including the case of a 13-year-old boy with preoperative concern for testicular malignancy.D. repensis becoming more relevant in central Europe. It should be considered in patients presenting with subcutaneous nodules in correlation with a travel history to endemic areas. Knowledge of this disease is essential for timely diagnosis and correct surgical treatment.As international traveling continues to grow and the natural habitat of mosquitoes expands,


EWSR1::CREM fusion are rare and challenging to treat. Pazopanib, a multi-tyrosine kinase inhibitor, is FDA-approved for advanced soft tissue sarcomas, but predictive biomarkers for its efficacy remain unidentified. We conducted a study on > 240,000 neoplasms submitted to Caris Life Sciences  to detect rearrangements using whole transcriptome sequencing. Two sarcoma-experienced, board-certified pathologists performed histological reviews, and treatment/outcome information was collected. Among the identified cases (n = 18), we observed a diverse range of sarcoma and other cancers, including an intracranial myxoid mesenchymal tumor, mesothelioma, hyalinizing clear cell carcinomas of the head and neck, clear cell sarcomas, and undifferentiated round cell sarcomas, as well as histologically malignant tumors with epithelioid morphology. Notably, two undifferentiated, metastatic, abdominal round cell sarcoma cases treated with pazopanib demonstrated significant sustained partial response and clinical benefit. To explore the genetic factors associated with the efficacy of pazopanib in these cases, next-generation sequencing and fluorescence in situ hybridization were analyzed for alterations in the tumors. The genomic analysis provided compelling evidence confirming the presence of EWSR1::CREM fusion in both cases, with no other pathogenic gene variants or copy number alterations detected. These cases demonstrate the potential of Pazopanib as a promising therapeutic option for patients with EWSR1::CREM fusion-positive soft tissue sarcomas, including metastatic undifferentiated round cell sarcomas. The sustained clinical benefit and partial responses observed in these cases warrant further research to validate these findings and explore the wider utility of Pazopanib in this rare and challenging subset of soft tissue sarcomas. Case studies: Case 1: A 49-year-old man presented with abdominal pain, weight loss, and chronic cough. A computed tomography (CT) of the chest, abdomen, and pelvis showed multiple lung nodules and masses and a right rectus mass that was biopsied and revealed an undifferentiated round cell sarcoma with a rare fusion EWSR1-CREM. No additional pathogenic gene variants or copy number alterations were detected. He received neoadjuvant chemotherapy with three cycles of Vincristine, Adriamycin, and Ifosfamide (VAI) and seven cycles of Vincristine/Irinotecan and Temodar (VIT). After cycle 7 of VIT, he had surgical resection of the abdominal mass and received radiation for lung metastasis. He completed 13 cycles of VIT after which he presented with progression of disease and switched to monotherapy with Pazopanib. At the time of this analysis he had stable disease for 28 months. Case 2: A 75-year-old woman presented with pelvic pain and new onset constipation. CT abdomen showed a large pelvic mass and intraperitoneal tumor spread. Exploratory laparotomy revealed a ruptured pelvic mass and a small bowel tumor. Both tumors were proved to be high-grade, poorly differentiated sarcoma. Genomic analysis demonstrated an EWSR1::CREM fusion but no other pathogenic gene variants or copy number alterations. She was treated initially for a primitive neuroectodermal tumor (PNET) with four cycles of Vincristine/Adriamycin/Cytoxan/Olaratumab but declined additional chemotherapy after progression. Two years later, she presented with recurrent abdominal mass and received one cycle of Temodar/Irinotecan, then she began Pozapanib and underwent palliative radiation to the entire pelvis. She has been on Pazopanib for 23 months with stable disease.Soft tissue sarcomas harboring  Sarcomas are rare tumors with an incidence of approximately five cases per 100,000. The 2020 World Health Organization classification recognizes more than 175 soft tissue and bone tumor entities .EWSR1) and a member of the TES transcription factors. Most cases are composed of uniform small round cells with scant, clear, or eosinophilic cytoplasm and, sometimes, it can present a higher grade of neuroectodermal differentiation    and has nd CREM) , intracrnd CREM) , clear cnd CREM) , hyalinind CREM) , and myxnd CREM) .EWSR1 status is not currently known to be predictive of targeted therapy, there is evidence that supports the role of multi-kinase inhibitors in treating sarcomas that harbor EWSR1 fusion (Pazopanib is a multi-tyrosine kinase inhibitor that has been approved by the FDA for the treatment of advanced soft tissue sarcomas (STSs) in patients who have previously received chemotherapy . Pazopan1 fusion .EWSR1::CREM rearrangements by whole transcriptome sequencing was performed for > 240,000 neoplasms submitted to Caris Life Sciences . Following the sequencing analysis, two board-certified pathologists (M.E. and A.R.) conducted a thorough histological review of the identified tumors. Available treatment and outcome information was obtained from the medical records.Detection of EWSR1::CREM fusion, encompassing various tumor types, including one intracranial myxoid mesenchymal tumor, one mesothelioma, four hyalinizing clear cell carcinomas of the head and neck, two clear cell sarcomas, two undifferentiated round cell sarcomas, and eight histologically malignant tumors with epithelioid morphology. Some of these cases were consistent with a recently reported disease entity in the medical literature known as “epithelioid neoplasm with a predilection for mesothelial-lined cavities.” However, some cases exhibited unexpected neuroendocrine differentiation and were located at uncommon sites, such as the kidney, uterus, and cervical spine. Additionally, three cases displayed features of squamous cell carcinoma. We identified two patients diagnosed with STSs that harbor the EWSR1::CREM fusion gene and have responded well to Pazopanib treatment. We aimed to investigate other possible EWSR1::CREM fusion alterations in STSs and their potential implications for the treatment of this type of cancer.Among the cases examined, 18 exhibited an A 49-year-old man with a past medical history of sarcoma of the right upper extremity in his mid-30s presented with 3 months of abdominal pain and unintentional 20 lb of weight loss over 3 months, despite normal appetite and productive cough for 8 months after multiple antibiotic treatments. Exact details of his first sarcoma diagnosis are unknown, but he received neoadjuvant chemotherapy followed by resection and adjuvant radiation. He had no oncologic follow-up after completing treatment for his first tumor. Physical examination revealed a right lower quadrant abdominal mass on palpation. Computed tomography (CT) chest, abdomen, and pelvis and fluorodeoxyglucose–positron emission tomography/CT (FDG PET/CT) showed multiple FDG-avid sites of disease, including multiple lung nodules, an 8-cm mass in the left upper lung lobe with direct extension to the mediastinum, and a 2.7-cm right rectus sheath mass  and exon 6 of CREM (transcript NM_001267562.1).A biopsy of the abdominal mass revealed a poorly differentiated malignant neoplasm, diagnosed as an unclassified round cell malignant neoplasm. The possibility of metastasis from the prior arm malignancy was considered. The lesion was composed of non-distinctive round cells with very limited amounts of eosinophilic cytoplasm and a high nuclear/cytoplasmic ratio with extensive necrosis. Immunohistochemical studies proved to be inconclusive. A month later, resection of the tumor allowed for the identification of an undifferentiated round cell sarcoma  and CD34 (a minority) and negative for DOG1, SMA, desmin, S100, HMB45, and inhibin. Full-thickness involvement of the small bowel wall was identified in the second lesion. Due to the anatomic location and CD117 expression, the possibility of a gastrointestinal stroma tumor was considered; however, morphology and lack of staining for DOG1 challenged this differential. Genomic analysis detected an EWSR1::CREM fusion was identified by genomic analysis. The patient received one cycle of Temodar/Irinotecan, then she began Pazopanib and underwent palliative radiation to the entire pelvis.The patient was treated for a PNET and completed four cycles of Vincristine/Adriamycin/Cytoxan/Olaratumab after which, he progressed and declined additional chemotherapy. Almost 2 years later, during follow-up, the patient complained of abdominal discomfort, pain, pressure, and bloating in the upper right abdomen and pelvis. A CT scan revealed a 6 cm × 6 cm ovoid soft tissue with multiple fluid densities within the pelvis. A PET/CT showed an ill-defined soft tissue density in the right mid-abdomen with mildly increased FDG uptake; as well as heterogeneous predominantly hypoattenuating lesions in the pelvis with heterogeneous mildly increased FDG uptake. A biopsy was taken which showed malignant round, epithelioid, and small spindle cells enmeshed in a focally myxoid stroma  reported five cases with stable disease and two with partial response  , are known to cause Ewing sarcoma and neuroectodermal tumors. EWSR1::CREM rearrangements are believed to lead to constitutive activation of CREM and subsequent dysregulation of oncogenes, such as BCL-2 family genes, EGFR1, and cyclins  for the publication of any potentially identifiable images or data included in this article.LC: Conducted a comprehensive literature review, collected and meticulously analyzed the patient data, and drafted the initial version of the manuscript. FC, ME, and AR: Conducted a thorough pathological review of the patients’ pathology specimens, contributed valuable feedback to the manuscript, and ensured the accuracy of the pathological data. FA: Reviewed the radiological images, interpreted and assessed the radiological response to treatment, and provided essential input to the manuscript. ME and AE: Interpreted the results of genetic analysis of the patient specimens, and provided critical feedback on the manuscript. JT: Designed and oversaw the study, provided expert guidance and supervision throughout the research, and contributed valuable feedback to the manuscript. All authors have carefully reviewed and approved the final version of the manuscript.


Remote cerebellar hemorrhage (RCH) is a rare complication following supratentorial craniotomies with unclear pathophysiology, predisposing factors, and clinical outcomes. This is a case of a 46-year-old female who presented to the emergency room with a complaint of severe headache associated with nausea. MRI studies demonstrated right frontal lesions consistent with low-grade glioma. She underwent a right frontal craniotomy, and the tumor was resected successfully. She developed a severe headache on postoperative day five, and CT scans showed ipsilateral cerebellar hematoma. She was managed conservatively and made a complete recovery within five days. Although rare, RCH requires prompt recognition, neurological monitoring, and management. Medical management and observation may be considered for patients without mass effect or acute hydrocephalus. It is well-documented that the occurrence of intracranial hemorrhages in the surgical area is a common risk associated with craniotomies, surgical evacuations, and resections, including those performed in the supratentorial region of the brain . ConversA 46-year-old female who has been a known case of controlled mass-induced seizure on levetiracetam for 20 years presented to our emergency room with a complaint of severe persistent headache-associated nausea and vomiting. The patient visited the emergency department for similar complaints three times over a period of six months. She was previously diagnosed with a brain space-occupying lesion in another institution where she was started on antiepileptic medications but refused surgical treatment. The neurological examination was unremarkable. Brain CT showed a right frontal intra-axial hypodense lesion with calcification associated with vasogenic edema Figure . Brain MA right frontal craniotomy and gross total resection of the lesion was carried out uneventfully. The ventricles were not breached during the surgery and its integrity was not compromised. Postoperatively, she was extubated in the operating theater, was vitally stable, and had no neurological deficits. Day one postoperative CT was unremarkable apart from expected postoperative changes following tumor resection, and the cerebellum was unremarkable. Histopathological examination of the surgical specimen revealed an isocitrate dehydrogenase (IDH)-mutant glioma with 1p19q codeletion on genetic studies consistent with oligodendroglioma (WHO 2).Postoperative MRI revealed no significant decompression of the supratentorial space with no invasion of the lateral ventricles Figure .Perioperative monitoring of blood pressure, coagulation, and neurological status were all normal. On postoperative day two, prophylactic subcutaneous enoxaparin was initiated. On the fifth day after surgery, the patient suddenly experienced a severe headache that was not associated with decreased level of consciousness, cerebellar signs, or seizures. During the patient's hospital stay, especially days two to five, there were no episodes of seizures or high blood pressure. Urgent brain CT scan revealed an intra-parenchymal hematoma in the right cerebellar area with surrounding cytotoxic edema with patent fourth ventricle and quadrigeminal cisterns Figure .Laboratory test results, including the coagulation profile, were normal. Conservative treatment with dexamethasone was initiated, and on the 10th postoperative day, the patient fully recovered and was discharged home.RCH is a rare complication associated with supratentorial craniotomies. It is characterized by intraparenchymal cerebellar hemorrhage distant from the surgery site . While RA systematic review of 209 patients conducted by Sturiale et al. (2016) found that around one-third of patients had asymptomatic RCH that was discovered incidentally in postoperative imaging. When symptomatic, patients exhibit a decreased level of consciousness and cerebellar signs, seizures, and in rare cases, coma . Our patThe risk factors of developing RCH are history of hypertension, coagulopathy, antiplatelet/anticoagulant use especially within a week before surgery, and perioperative complications such as seizures and hydrocephalus . None ofThe symptoms of RCH may vary, and the condition is often diagnosed by neuroimaging. Patients diagnosed with RCH demonstrate distinct radiological characteristics that distinguish this form of intracerebral hemorrhage from others. These features manifest as a pattern of alternating hyperdense and hypodense curvilinear structures with irregular stripes, which resemble the distinctive coat of a zebra and have consequently been coined the "Zebra-sign" . The occSeveral theories have been proposed to explain the development of this complication, including rapid loss of cerebrospinal fluid (CSF), resulting in shearing forces and tearing of the cerebellar veins . Konig eManagement of RCH depends on the clinical status and radiological findings. RCH cases are usually benign, and the majority of patients improve with observation or medical management. However, patients must be carefully monitored for any potential deterioration. Generally, older patients were correlated with poorer outcomes, while the asymptomatic presentation was associated with a favorable prognosis . In someDespite being a rare complication following supratentorial craniotomy and resection of space-occupying lesions, remote cerebellar hemorrhage necessitates prompt recognition and neurological monitoring for any signs of further deterioration. Medical management and observation should be initiated for patients without mass effect or acute hydrocephalus. Surgical decompression is to be considered when the patient's level of consciousness is decreased due to posterior fossa tightness. In cases of acute hydrocephalus, external or ventriculoperitoneal shunting may be beneficial. Timely decision-making and management are crucial to the best possible outcome. Further research is necessary to better understand the mechanisms and risk factors associated with remote hemorrhages following brain tumor resection.


Biliary pathologies are common causes of acute pancreatitis, including gallbladder cholesterolosis and gallstone pancreatitis. Nevertheless, after these two pathologies have been excluded, a broader differential and less common etiologies must be considered. Here we report an 18-year-old female with preliminary diagnosis of gallstone pancreatitis who underwent cholecystectomy and intraoperative cholangiogram resulting in uneventful recovery and resolution of symptoms. She returned three times within the month with symptoms of acute pancreatitis. We discussed alternative etiologies of her pancreatitis after exclusion of other causes with extensive imaging and laboratory evaluation. The annual incidence of acute pancreatitis worldwide is increasing due to increased prevalence of gallstones and obesity. Gallstones account for 40-70% of all cases, especially in female patients as they are at higher risk for cholelithiasis . AnotherOn 08/03/23, an 18-year-old female presented to the emergency department (ED) with moderate to severe pain in the epigastric and right upper quadrant for the past week. Her last meal before the pain consisted of yogurt and chicken salad. She had no significant past medical history and did not drink alcohol or use tobacco products. Her body mass index was 33.3. The only medication was a birth control patch. At ED admission, the patient had stable vital signs as follows: blood pressure 131/81, pulse rate 71 per minute, respiratory rate of 15, temperature of 97.5 °F (36.4 °C), oxygen saturation of 96%. Laboratory results were significant for elevated lipase and leukocytosis but the remainder, including hepatic panel, were normal  of abdomen and pelvis demonstrated mild edematous changes in the tail of the pancreas, suggesting pancreatitis Figure . Her lipThis patient’s recurrent pancreatitis post-op challenges the primary diagnosis of cholecystic origin. In many patients with acute idiopathic pancreatitis, cholecystectomy usually results in the resolution of symptoms. According to a recent systematic review and meta-analysis, the recurrence rate of pancreatitis is lower after cholecystectomy in patients with idiopathic acute pancreatitis . HoweverThe Milwaukee Classification describes biliary and pancreatic as two main categories of SOD with three subtypes within each category based on clinical presentation, laboratory findings, and imaging . BiliaryPost-cholecystectomy idiopathic pancreatitis is a rare presentation that should raise red flags in clinical settings. It should challenge clinicians to reconsider the primary clinical diagnosis prior to the surgery, and prompt further workup for less common etiologies. Both the negative results for autoimmune pancreatitis and the minimum lipase value recorded after IOC peripherally suggest a sphincter pathology. Furthermore, her refractory pain regardless of dietary changes and NSAID use also corroborates with the spasmic pressure pain due to SOD. The normal hepatic panel throughout her stay makes the more common biliary SOD less likely. Overall, the clinical course, laboratory results, and imaging results for this patient with acute recurrent pancreatitis suggest pancreatic type II SOD as the probable etiology.


Actinomyces spp is not uncommon, particularly among intrauterine device users. Pelvic actinomycosis is an extremely rare disease. The clinical picture can resemble an advanced ovarian malignancy. We report a case of pelvic actinomycosis mimicking ovarian malignancy diagnosed postoperatively. Preoperative diagnosis is possible if there is a high index of suspicion, obviating extensive surgery and preserving fertility, since long term antibiotic treatment can be completely effective. Pelvic actinomycosis should be included in the differential diagnosis of women presenting a pelvic mass, especially if there is intrauterine device use history.Asymptomatic female genital tract colonization with  Actinomyces spp, most commonly Actinomyces israelii. Actinomyces are commensal organisms that colonize the human oral cavity, gastrointestinal and genital tract.Actinomyces are normally unable to cross the mucosal barrier. Tissue injury, such as trauma, surgery, or foreign body, is required for progression from colonization to clinical infection. Once the mucosal barrier has been breached, the bacteria usually spreads by continuity (lymphatic and hematogenous spread are uncommon), invading surrounding tissues and originating abscesses and sinus tracts.Pelvic actinomycosis is a rare, chronic, suppurative and granulomatous disease caused by the anaerobic Gram-positive bacteria Abdominopelvic actinomycosis comprises ∼ 20% of reported cases of actinomycosis.The ability of this disease to mimic pelvic malignancy has been previously presented in several case reports and case series.Medical treatment alone with penicillin is highly effective and can successfully eliminate pelvic actinomycosis, avoiding extensive extirpative surgery and preserving fertility.A 47-year-old woman was referred for gynecological evaluation by her attending gastroenterologist, after a pelvic mass was found on a computed tomography (CT) scan.The patient was born in Brazil and had been living in Portugal for the past 26 years. Her medical history was unremarkable. She used a copper IUD for 6 years and she had it removed 4 months before. Her last pap smear, 2 years earlier, showed no abnormalities.A concentric infiltrative rectal lesion was noted in a screening colonoscopy she had, given her family history of colorectal cancer. Biopsies of this lesion were inconclusive, without dysplasia. A CT of the thorax, abdomen and pelvis was ordered and revealed a large solid heterogeneous pelvic mass, left hydronephrosis and iliac lymph node enlargement .The patient had no complaints. At physical examination, a hard, tender, palpable mass in the posterior cul-de-sac was noted; no other abnormalities were detected.A pap smear was obtained and the results were normal. Laboratory studies only revealed mild normocytic anemia . The white blood cell count, erythrocyte sedimentation rate and C-reactive protein were normal; serum creatinine was normal (0.60 mg/dL). Tumor markers were also within the normal range .Pelvic magnetic resonance showed a heterogeneous, mixed, retrouterine mass, probably of left adnexal origin, measuring 6 × 5 × 5 cm . The masA malignant ovarian tumor was the leading diagnostic hypothesis. Alternative diagnoses considered were a primitive colorectal tumor with adnexal metastasis and deeply infiltrating endometriosis.Definite diagnosis required histologic sampling of the mass. The patient had completed child-bearing and had no desire of preserving fertility. A multidisciplinary pelvic oncology team discussed the case. Given the ureteral and bowel compression, it was thought the mass required surgical resection, regardless of its etiology. A laparotomic approach was chosen, given the degree of suspicion of malignancy, the predominantly solid nature of the mass, its dimensions, and the level of expertise in laparoscopy at our center.The surgical plan was discussed with the patient, who gave informed consent for left adnexectomy with intraoperative frozen section and a possible complete surgical staging procedure for ovarian cancer and even segmental bowel resection.A left ureteric stent was placed prior to surgical intervention.en bloc resection of the described mass were performed. The resection was incomplete due to the lack of cleavage plane with the bony pelvis. The extemporaneous examination favored a benign inflammatory etiology and the surgery was concluded.On exploratory laparotomy, a hard, left adnexal mass with ∼ 6 cm, extending to the rectum, sacrum and left pelvic wall was found. Given the infiltrative nature of the mass and the lack of cleavage planes with the nearby structures, and since the patient had no desire of preserving fertility, it was decided to complete total hysterectomy and bilateral salpingo-oophorectomy. Peritoneal lavage, total hysterectomy, right adnexectomy and Final histopathological examination established a diagnosis of left tubo-ovarian actinomycosis, with active chronic inflammation and abscess formation.Intravenous penicillin (5 million units every 6 hours) was administered for 4 weeks, followed by oral doxycycline for 12 months.A follow-up CT of the abdomen and pelvis, 1 month after completing the penicillin course of treatment, showed no signs of the disease.The patient had follow-up appointments about every 3 months for over 1 year, either with her attending gynecologic oncologist or infectiology specialist. During this time, she remained asymptomatic, except for troublesome vasomotor symptoms. Transdermal estrogen was prescribed with adequate relief.Actinomyces in IUD users must be differentiated from clinically relevant pelvic actinomycotic infection.Despite the association between IUD use and pelvic actinomycosis, asymptomatic genital tract colonization by Actinomyces exists in normal oral and gastrointestinal flora. Female genital tract colonization is also not uncommon. The incidence of Actinomyces-like organisms was 0.26% in a study of more than 20,000 pap smears.Actinomyces-like organisms in cervical smears were IUD users (81%), with 60% having a copper IUD and 31% a levonorgestrel-releasing intrauterine system (LNG-IUS).Actinomyces-like organisms can be found in cervical smears in up to 7% of IUD users.Actinomyces, and only half the diagnosis made through pap smears are actually culture positive.Actinomyces genital colonization; however, it is probably exceedingly low.Actinomyces on a Pap smear is considered incidental and the asymptomatic patient does not require antimicrobial treatment or removal of the IUD.It is impossible to quantify the risk of developing serious pelvic infection in IUD users with The rate of actinomyces-like organisms in cervical smears is lower with the more recent LNG-IUS than with the copper IUD.In this case report, the patient had a copper IUD in place for 6 years, which had been removed 4 months before presentation. Although the development of pelvic actinomycotic abscesses in women with IUDs is exceptionally rare, the diagnosis should be considered in women of reproductive age with a pelvic mass, especially in those with an IUD in place or recently removed.Actinomyces-like organisms.Actinomyces in two pap smears collected 2 years apart.Among women with pelvic actinomycotic abscesses, only 50% had a previous pap smear positive for Given its rarity and lack of distinct clinical features, pelvic actinomycosis is a difficult diagnosis. Common symptoms include lower abdominal or pelvic pain, abnormal uterine bleeding or discharge, a palpable abdominopelvic mass, fatigue, weight loss, fever and symptoms related to bowel obstruction or obstructive uropathy. There may be anemia, mild leukocytosis, elevated C-reactive protein and elevated erythrocyte sedimentation rate.Several aspects make this case atypical in its presentation. The patient had no symptoms, and the pelvic actinomycotic abscess was incidentally discovered because a rectal infiltrative lesion was found during a screening colonoscopy. Also, iliac lymph node enlargement was reported on CT. Local or regional lymphadenopathy is unusual in actinomycosis.Pelvic actinomycosis clinically mimics ovarian cancer and other diseases, such as tuberculosis, pelvic inflammatory disease, lymphoma, inflammatory bowel disease, diverticulitis or endometriosis.Correct nonsurgical diagnosis is possible by image guided biopsy or laparoscopic biopsy and prolonged antibiotic therapy can avoid extensive extirpative surgery and preserve fertility.A diagnosis of advanced ovarian cancer is often assumed and these patients are subjected to unnecessary exploratory laparotomy.Actinomyces.Actinomyces are fastidious organisms, and less than 50% of suspected cases are confirmed by this method.The final diagnosis is based on the recognition of actinomycotic sulfur granules on histology and/or on cultural identification of High-dose prolonged therapy with penicillin has been the treatment of choice for actinomycosis. Traditionally, intravenous penicillin G at a dose of 18 to 24 million units a day is administered for 2 to 6 weeks, followed by oral penicillin V (2–4 g/day) for 6 to 12 months. Doxycycline, minocycline, clindamycin and erythromycin are considered reasonable alternatives for patients allergic to penicillin. Medical treatment can be completely effective.Surgical treatment alone is not curative but may be a useful adjunctive in selected cases. It also may be necessary if malignancy cannot be excluded.Actinomyces-like organisms on a pap smear does not require antimicrobial treatment or IUD removal.Pelvic actinomycosis should be included in the differential diagnosis of pelvic masses suspicious for malignancy. Doctors should be especially aware of this disease when there is a history of IUD use. Non-surgical diagnosis of pelvic actinomycosis is possible, and antibiotic treatment can be completely effective, avoiding extensive surgery and preserving fertility. Pelvic actinomycosis is very rare; however, asymptomatic female genital colonization by this organism is not uncommon. The incidental finding of


Bilateral adrenal myelolipoma is rare in clinics and patients with disorders of sex development (DSDs). One case was reported in our center. A 45-year-old patient was admitted to the hospital after discovering a left abdominal mass for more than a year and worsening abdominal pain for 18 days. An imaging examination showed bilateral adrenal masses. Physical examination showed clitoris hypertrophy with patelliform changes, thick and dense pubic hair, normal development of bilateral labia majora without labia minora, and urethral opening. After the relevant preoperative examinations, bilateral adrenal mass resection was performed under general anesthesia. The postoperative pathology confirmed adrenal myelolipoma. The incision healed well without recurrence over 10 years after the operation. Her enlarged clitoris decreased in size. This case report has a detailed diagnosis and treatment process and sufficient examination results. It can provide a reference for diagnosing and treating patients with bilateral adrenal myelolipoma and DSD and reduce the risk of misdiagnosis and mistreatment. Myelolipoma is a rare non-functional benign tumor formed by the proliferation of mature adipose tissue and hematopoietic components of bone marrow. It was first described histologically by Gierke in 1905 and named myelolipoma by Oberling in 1929 . BilaterThe patient was 45 years old, and the social gender was female. The patient was admitted to the hospital after discovering a left abdominal mass for over a year and aggravation of abdominal pain for 18 days. In January 2008, the patient developed abdominal pain and discomfort without obvious causes. A left abdominal mass, about 4 cm × 3.5 cm × 3 cm in size, was found in the local hospital. In January 2009, the patient touched the left abdomen while bathing and felt that the mass was enlarged and hard, and abdominal pain recurred simultaneously. She was admitted to our hospital with a left abdominal mass of unknown etiology in February 2009. The patient had no fever, night sweats, jaundice, or emaciation. She had no menses since puberty. There was no family history.Physical examination revealed a temperature of 37.2°C, respiration of 18 beats per minute, pulse of 72 beats per minute, and blood pressure of 120/80 mmHg. The patient had a body weight of 52 kg and a male face with visible whiskers and throat segments and a coarse, deep voice. The axillary hair was thick and dense, the chest was symmetrical without deformity, the breasts were not developed, and a physical examination of the heart and lungs showed no positive signs. The abdomen was soft with no palpable liver. However, a large mass was palpable in the left abdomen with a slightly hard texture and poor mobility, and there was no percussion pain in both renal region. The vulva showed hermaphroditism, thick and dense pubic hair, normal development of bilateral labia majora, no labia minora, hypertrophy of the clitoris like a short penis (4 cm long), no opening at the top, the urethral opening in the vestibule below the clitoris, and no vagina. On digital rectal examination, there was no palpable uterus or prostate-like tissue, no palpable mass in the pelvic cavity, and no testis-like tissue in the inguinal canal area: scoliosis deformity and free movement of limbs.9/L, neutrophils of 74.7%, lymphocytes decreased by 13.9%, and monocytes increased by 10.5%. The red blood cell count was 5.30 × 1012/L, and the hemoglobin was 177 g/L. Total bilirubin, direct bilirubin, and indirect bilirubin increased to 30, 11.3, and 18.7 μmol/L, respectively. Total cholesterol decreased to 2.55 mmol/L, and high-density lipoprotein decreased to 0.95 mmol/L. Other parameters, such as blood glucose and renal function, were normal. Tumor markers such as alpha-fetoprotein, human chorionic gonadotropin, neuron-specific enolase, and carcinoembryonic antigen were negative. Karyotype analysis of the patient’s peripheral blood showed that the patient was 46, XX.Abdominal CT examination showed a huge mixed-density lesion (18 cm × 15 cm) in the left retroperitoneal space, including many irregular fat density shadows. The upper boundary of the mass was below the diaphragm, and the lower boundary was at the level of the iliac crest. The pancreas, stomach, and spleen were all compressed and displaced anteriorly, and the left kidney was compressed into the pelvis. A mixed-density shadow of 8 cm × 4 cm with clear borders was seen above the right kidney. On enhancement, the parenchymal portion of the left retroperitoneal mass showed variable enhancement that appeared to be encapsulated , dark, and hard in texture. The mass was dissected carefully to avoid injury to the renal vein. The huge mass was completely removed, and the other side was about 9 cm × 5 cm removed. A part of the bilateral mass after complete resection was sent for intraoperative rapid frozen section diagnosis. Intraoperative frozen report: The tumor was derived from adipose tissue and tended to be benign. Lymph node dissection was not performed. After sufficient hemostasis of the operative field, a drainage tube was placed, and the abdominal incision was sutured layer by layer. The intraoperative blood loss was about 500 mL. The operation time was 3 h and 15 min. The operation was uneventful, and anesthesia was stable without a blood transfusion. The patient returned to the ward safely after the operation. The drainage tube was removed 7 days after the operation, and the incision healed well 14 days after the suture was removed. The patient was discharged.Pathological examination: gross observation: The left side was a yellow-red or dark-red mass, weighing 2.3 kg, measuring 24 cm × 18 cm × 10 cm, with a complete capsule, solid cut surface, and pale yellow fat mixed with reddish-brown , and hyperparathyroidism . CAH is There are many studies on the nature of myelolipomas. Bishop et al.  found thMyelolipoma is generally asymptomatic, with a volume of less than 5 cm and rarely larger than 10 cm . The larIn conclusion, myelolipoma is rare in clinical practice, and it is rare to present a huge mass in both adrenal glands with DSD. Adrenal myelolipoma is a benign non-secretory tumor. The tumor was named for the presence of mature adipocytes and bone marrow cells. The disease may result from abnormal cortical reticular endothelial cell metaplasia development or aberrant embryonic residues. However, patients with adrenal myelolipoma that produce compression symptoms such as hypertension need surgical resection of the tumor to relieve the symptoms. Adrenal myelolipoma is considered a benign tumor without secretory function, and postoperative chemoradiotherapy and endocrine therapy are not needed. Patients with adrenal myelolipoma complicated with disorders of sexual development can be followed up after resection of adrenal myelolipoma before oculoplastic surgery, and some patients with disorders of sexual development can spontaneously relieve the abnormal manifestations of external genitalia.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by the Ethical Committee of Kunming Children’s Hospital (2022-12-001-K01). This study is by the relevant guidelines and regulations. The data in this study were obtained from this patient and his legal guardian. Written informed consent was obtained from the patient’s parents.CZ designed the study. CZ, NL, and ZY collected and analyzed the data. CZ drafted the initial manuscript. CZ revised the article critically. CZ, ZY, NL, BY, and YX reviewed and edited the article. NL and CZ are co-first authors. All authors approved the final manuscript.


Lower urinary tract symptoms (LUTS) are a relevant problem in the pediatric population, having a very high prevalence. Diurnal incontinence and nocturnal enuresis are surely the most frequent symptoms, presenting, respectively, in up to 30% of school-age children and up to 10% of children between 6 and 7 years. Stypsis is the most common comorbidity, and it must be considered in the management of LUTS; indeed, the treatment of constipation is curative in most cases for both incontinence and enuresis. The presence or absence of diurnal symptoms in nocturnal enuresis and urgency in diurnal incontinence helps in the differential diagnosis. Urotherapy is always the first-line treatment, while oxybutynin and desmopressin (where appropriate) may help if the first-line treatment is unsuccessful. It is essential to identify conditions that are potentially dangerous for kidney and urinary tract well-being, for which LUTS can be the first manifestation. Starting from a series of clinical scenarios, we will underline the diagnostic clues behind LUTS in children and we will summarize clinical and surgical approaches for the proper management of these conditions. Symptoms deriving from the lower urinary tract are grouped under the umbrella term “lower urinary tract symptoms” (LUTS). LUTS are classified by the International Children’s Continence Society (ICCS) according to their relationship with the voiding or storage phase of the bladder cycle or a combination of both in changing severity. Mainly, the conditions are classified into either overactive bladder (OAB) or dysfunctional voiding. Storage symptoms are represented by increased (≥8 times/day) or decreased (≤3 times/day) number of diurnal voiding, daytime continuous or intermittent incontinence, enuresis, urgency, and nocturia. Voiding symptoms are classified as hesitancy, straining, weak stream, and intermittency .LUTS are very frequent in the pediatric population, with daytime incontinence, defined as an uncontrollable leakage of urine, present in up to 30% of school-age children. Holding manoeuvres and urgency, which is a sudden and unexpected experience of an immediate need to void, are also highly prevalent . Common Comorbidities are a relevant problem in these patients. ICCS suggested a list of comorbidities such as constipation, encopresis, urinary tract infection (UTI), vesicoureteral reflux (VUR), and neuropsychiatric conditions  that must be considered when studying LUTS properly. Stypsis is surely the commonest and most important comorbidity: up to 30% of children affected by stypsis have diurnal urinary incontinence and/or enuresis. On the other hand, proper stypsis management alone can resolve 90% of diurnal urinary incontinence cases and 60% of enuresis cases . HoldingLUTS diagnosis is not always easy to reach and so the treatment could be delayed. Firstly, LUTS are often underestimated by primary care paediatricians who tend to wait for a spontaneous resolution of the condition . SecondlStarting from a series of “illustrative” case reports, we will underline the diagnostic issues and the diagnostic clues behind LUTS in children to help paediatricians in orienting toward the differential diagnosis and the proper management of these conditions.An 8-year-old girl was referred to our outpatient clinic for urgency, urge incontinence, and increased voiding frequency (12–14 times/day) in the last year. Before that, no urinary symptoms were described. Nocturnal and diurnal continence was attained at 2.5 years old and no UTI was reported by the mother. No malformations at the external genitalia examination were found. Urine analysis was normal. At the kidney and urinary tract ultrasound (KUS), both kidneys and bladder were normal and no post-void residual volume was found. The uroflowmetry revealed a tower-shaped curve. This clinical picture was suggestive of OAB. Standard urotherapy was the first line of treatment and an anticholinergic agent (oxybutynin) helped in the later management.A 7-year-old boy came to our outpatient clinic with increased daytime voiding frequency (more than 15–20 times) presenting in the last 5 months. No urinary urgency, nocturia, daytime incontinence, or nocturnal enuresis were reported. Stypsis was absent and urinalysis was normal. The clinical examination was unremarkable. The bladder voiding diary revealed 37 micturitions/day, with a volume void of each micturition between 10 and 40 mL. Based on this clinical presentation, the diagnosis of extraordinary daytime-only urinary frequency (EDOUF) was made. We reassured the parents about a benign prognosis and spontaneous resolution .A 4-year-old girl was observed for diurnal and nocturnal incontinence without urgency. The urinalysis was normal. The physical examination of external genitalia revealed a bifid clitoris, hypoplasia of minora labia, and patulous urethra due to a dorsal urethral defect. The diagnosis of female epispadias was found. Cystography was normal and the surgical correction of the defects determined the disappearance of the urinary incontinence . At the A 6-year-old girl presented with a history of daytime incontinence with increased daytime voiding frequency and enuresis. She also reported recurrent febrile UTIs since she was 18 months old. KUS showed bilateral hydronephrosis, with an anteroposterior diameter of the right pelvis of 21 millimetres and the left pelvis of 24 millimetres. The bladder wall was irregular and 6 millimetres thick (bladder fully filled). Moreover, she reported being styptic since she was 2 years old and recurrent nonfebrile UTIs in the last 6 months (about 3 episodes). Uroflowmetry revealed that a “staccato” pattern was present. An antibiotics prophylaxis with amoxicillin and a bowel regulation strategy (stool softeners plus lifestyle modification) were started. The girl was re-evaluated after 3 months, with normalizations of the symptoms, KUS, and uroflowmetry. In this case, the diagnosis was BBD, and all of the symptoms disappeared with proper urotherapy and stypsis management.An 8-year-old girl was observed for persistent urinary incontinence without urgency. She reached continence at 2.5 years old. At the physical examination, the genitalia were normal, but urine dripping was seen during the ostium vaginalis exploration. In the suspicion of an ectopic ureter, a KUS was performed, and a right kidney duplication with a megaureter of 8 mm of the upper pole was found. The Tc-99m MAG3 kidney scintigraphy revealed absent function (6%) of the right upper pole. In addition, we made a methylene blue test consisting of filling the vesical bladder with blue-coloured NaCl 0.9% by vesical catheterization. In this test, we found vaginal non-blue urine dropping, confirming that this dropping was derived from an ectopic ureter in the vagina and not from the vesical bladder. During cystoscopy, the ureteral orifice of the ureter of the upper pole was not found. The nephroureterectomy of the upper pole resolved the incontinence of the patient.An accurate anamnesis should be taken, focusing on symptoms reported by the child and confirmed by the caregiver. The paediatrician should investigate voiding patterns, frequency, bowel habits, fluid intake, and posturing taken during micturition. Moreover, a medical history of patients and relatives regarding nephro-urological diseases and other problems  should be considered. Asking for a history of constipation and recurrent UTIs should be the first step in the management of any urinary symptoms.Physical examination is essential and sometimes enough to make a diagnosis. Any anomalies should be noted, including ear anomalies, which can be associated with urinary tract malformations . AbdomenA urine analysis test is mandatory in excluding both mellitus (glycosuria), diabetes insipidus (low urinary density), and UTI (leukocyturia and/or nitrituria).At first evaluation, it should always be asked that a bladder diary be completed by the caretaker or patient. ICCS recommends at least a 48 h daytime frequency and volume chart to well evaluate LUTS . HoweverKUS is a noninvasive and useful investigation tool that helps physicians in cases of kidney abnormalities, such as urinary tract dilatations or kidney duplications. Bladder thickness in healthy children should not overtake 3 and 6 millimetres in a full and empty bladder, respectively . BladderUroflowmetry is functional for a correct diagnosis. It is considered a noninvasive urodynamic test. Therefore, it is useful in approaching children with urinary symptoms. It consists in letting a toilet-trained child void their bladder into a toilet connected to a sensor that measures urine flow, and it is always associated with a residual postvoid volume measurement at ultrasonography. All of the adaptions possible for children should be taken to let them void their bladder in a position that is normal for them . UroflowFinally, cystometry is surely the most definitive test to diagnose voiding dysfunction and outlet obstruction. It allows paediatricians to measure bladder compliance during the filling phase, calculated by dividing the volume change by the change in detrusor pressure. Both transurethral and suprapubic routes can be used. In any case, cystometry is not mandatory, and most cases are diagnosed with uroflowmetry and KUS association. Indeed, cystometry is associated with adverse effects, e.g., UTI, macroscopic haematuria, and urinary retention in up to 20% of cases .Generally, after the treatment of a possible underlying UTI, the first-line approach for any patient with LUTS is standard urotherapy. It is defined as a conservative-based therapy and treatment for LUTS that rehabilitates the low urinary tract . It consIn the case that standard urotherapy is unsuccessful, a specific urotherapy is the method of choice. An example is “biofeedback”, consisting in letting a child gain greater awareness of the pelvic floor and sphincter muscles by using external instruments . Up to 8A postponing micturition exercise, as in clinical scenario 2, could be used in patients with EDUOF diagnosis for which effectiveness in 98.1% of patients who improved or normalized their voiding frequency has been shown .In patients with MNE, standard urotherapy is more effective than spontaneous cure by a rate of 15% per year . Robson MNE should be treated with a combination of urotherapy and desmopressin or alarm therapy. The latter consists in putting a sensor in the night clothes, which gives an auditory signal that wakes the child when it gets wet. The alarm should be preferred in children with a bladder capacity smaller than expected, who are likely to be resistant to desmopressin. It should be worn every night, and treatment should be continued until the patient is dry for 14 consecutive days or 2–3 months . This ap2 and M3 receptors, which are predominantly expressed by bladder smooth muscle. Oxybutynin, at a dosage range from 0.3 to 0.5 mg/kg/day with a top dose of 10 mg [In the case of OAB, urotherapy is still the first choice . Antichoof 10 mg , is prefof 10 mg . Moreoveof 10 mg .In case of unresponsiveness, transcutaneous electrical nerve stimulation (TENS) could be considered. It consists of inhibiting the presynaptic afferent neurons carrying impulses from the bladder by stimulating the nerves of the peripheral segmental dermatome . FinallyIn conclusion, BBD and recurrent cystitis should be always excluded and eventually treated, as in scenario 4. In the case of the persistence of LUTS, a diagnosis revaluation with deeper diagnostic tools is required.Clinical scenarios 3 and 5 represent examples in which a clinical approach is not enough. In the case of female epispadias, an accurate genitalia examination is sufficient, and early surgical intervention is fundamental to let the patient achieve continence and preserve the urinary tract. An ectopic ureter is a more complex condition, and the diagnosis could not be as immediate as epispadias. The term “ectopic ureter” describes a ureter that inserts at or distal to the bladder neck, and it is more frequent in females . FrequenA surgical approach is needed in the case of posterior urethral valves (PUV), which are the most common congenital cause of lower urinary tract obstruction. PUV is usually diagnosed during routine prenatal ultrasounds with the detection of hydronephrosis, a thick-walled bladder, and a keyhole sign in the bladder neck of males (dilated prostatic urethra) . In the Proper management, with rapid diagnosis and treatment, is critical in children with LUTS because of the negative influence that LUTS can have on the quality of life. Both enuresis and daytime incontinence affect children and their families socially, behaviourally, and emotionally, with a risk of social isolation, peer conflict, and classroom challenges. Moreover, LUTS affect the individual in terms of development, self-esteem, and performance, as well as the relationship within the family, causing a high level of stress. Rangel et al. concluded that enuretic children have a low quality of life compared to non-enuretic children, without any differences in gender or age .Additionally, it is known that children with both OABs have a prevalence of VUR between 14% and 47%. On the same line, the treatment of overactivity has a positive effect on the resolution and downgrading of VUR .Moreover, the chronic obstruction caused by PUV can lead to hypertrophy of the bladder wall and detrusor muscle, with changes in bladder compliance. The consequent increment in intravesical pressure can cause VUR, and, thus, recurrent infection and progressively impaired renal function .Finally, dysfunctional voiding can present indistinguishably from the classical neurogenic bladder, and such patients may encounter bilateral hydronephrosis and renal failure . Both coLUTS are very common conditions in the pediatric population. They are usually underestimated and their diagnosis is not always immediate, with a frequent need to reevaluate patients. Many diagnostic tools are available to help clinicians orientate through clinical issues of LUTS. A clinical approach starting with urotherapy is usually sufficient for the management, while a few conditions need a surgical approach. In brief, urinary incontinence should be divided into nocturnal and diurnal incontinence. In the former, the co-presence of diurnal symptoms makes for a diagnosis of NMNE, while their absence makes for a diagnosis of MNE. In the case of diurnal incontinence, the presence or absence of urgency should be considered when making a differential diagnosis between OAB and other conditions that could require surgical intervention, such as an ectopic ureter. Medications, such as oxybutynin (in the case of NMNE or OAB) and desmopressin (MNE), are mostly used in the case of unsuccessful urotherapy.