Early-stage colorectal cancer (CRC) is often treated endoscopically, but additional surgical resection may be considered depending on pathological findings.A 73-year-old man was found to have early-stage sigmoid colon cancer by colonoscopy during a medical examination, and endoscopic mucosal resection (EMR) was performed. The lesion was a 7-mm-sized sessile polyp, and the pathological diagnosis was well-differentiated tubular adenocarcinoma, pT1 , with no lymphovascular invasion, low budding grade, and negative horizontal and vertical margins. Therefore, the patient was observed without postoperative treatment. One year later, a computed tomography (CT) scan showed multiple liver metastases. After five courses of preoperative chemotherapy with folinic acid, 5-fluorouracil and oxaliplatin (FOLFOX) and panitumumab, liver metastases were reduced. The patient underwent extended right hepatic lobectomy. The pathological finding was well-to-moderately differentiated tubular adenocarcinoma, and immunohistochemistry findings were consistent with liver metastases from sigmoid colon cancer. Postoperatively, the patient received five courses adjuvant chemotherapy with FOLFOX. Although the patient had been recurrence-free for 5 years after liver resection, a CT scan revealed a nodular lesion in the sigmoid mesentery. Positron emission tomography (PET) showed abnormal accumulation in the same lesion. Therefore, the mesenteric nodules diagnosed as lymph metastasis and recurrence of sigmoid colon cancer and performed laparoscopic sigmoid colon resection with lymph node dissection. The pathological findings showed that the recurrent lesion in the mesentery formed a nodular infiltrate with venous, lymphatic, and neural invasion, but lymph node structures were not found, and it was assumed to be metastasis or recurrence due to lymphovascular invasion. The pathologic specimen of the sigmoid colon had no neoplastic lesions, which are considered to be a local recurrence on the mucosal surface. After sigmoid colectomy, adjuvant chemotherapy with CapeOX was conducted, and the patient has been recurrence-free for 13 months at present.Even early-stage CRCs that have no pathological indications for additional resection have risks of metastases and recurrences, and we may need to consider that the criteria for additional resection should not be limited to pathological findings alone. Early-stage colorectal cancer (CRC) is often treated endoscopically, but additional surgical resection may be considered depending on pathological findings. Here, we report a case of a patient with early-stage CRC who did not meet the criteria for additional resection after endoscopic treatment but developed liver and mesenteric metastasis at a later date.A 73-year-old man was found to have early-stage sigmoid colon cancer by colonoscopy during a medical examination. Colonoscopy revealed a 7-mm-sized sessile and protuberant lesion in sigmoid colon; after observation with indigocarmine spray, we diagnosed early-stage sigmoid colon cancer Fig. , and endn = 104) in patients with CRC [Early-stage CRCs are divided into intramucosal (Tis) carcinomas and submucosal T1) carcinomas. The overall recurrence rate is approximately 1% for SM carcinomas in Stage I cases  carcinom–4 and liwith CRC . In the Regarding recurrence at a distant period, recurrence over 5 years after radical resection for early-stage CRC is extremely rare, and lymph node recurrence after 5 years from the last surgery for stage I CRC is estimated to be less than 0.08% of all cases after curative resections . One conThe course of the current case suggests that there are lesions that escape from chemotherapy without being completely obliterated and that they may be the cause of late recurrence. In other words, chemotherapy has limitations. Although the pathological diagnosis of our case did not meet the criteria for additional resection, the aforementioned reports –10 suggeIn addition, in this case, although there were no pathological risk factors for surgical treatment just after EMR for primary resection, liver and mesenteric metastasis still occurred. Although we cannot completely rule out the possibility that the finding of lymphovascular invasion was overlooked, despite multiple pathological examinations, we found no evidence of lymphovascular invasion. In other words, subsequent surgical treatments could have been avoided if residual tumor cells could have been predicted and additional sigmoid colon resection could have been performed after EMR or at the same time as liver resection. Therefore, we need methods other than pathological factors that can correctly evaluate the risk of metastasis. The tumor marker CEA may be used as a complement for risk assessment but has limited specificity and sensitivity . RecentlWe have experienced a case of liver metastasis and late mesenteric recurrence after EMR for early-stage sigmoid colon cancer. It is necessary to keep in mind that such a course can occur even in patients with curative early-stage cancer after EMR.We reported a rare case of early-stage sigmoid cancer that caused liver metastasis and mesenteric metastasis at a distant stage occurring 7 years after the first surgery. Even early-stage CRCs that have no pathological findings for additional resection have risks of metastases and recurrences, and we may need to consider that the criteria for additional resection should not be limited to pathological findings alone.


Colorectal carcinoma (CRC) should be suspected in pediatric patients with a bowel obstruction in an emergency setting. Evidence‐based surgical management with chemotherapy is crucial to prevent adverse outcomes.CRC should be suspected in pediatric patients presenting to the emergency with unspecified abdominal pain. An erect X‐ray abdomen and a colonoscopy should be initial diagnostic tests. If colonoscopy raises suspicion of CRC, a biopsy during colonoscopy is indicated. A computed tomography scan of the chest, abdomen, and pelvis will also aid in diagnosis, staging, and planning intervention. In advanced cases, the intervention includes proximal diversion, bypass, and bowel resection with anastomosis. Sometimes postoperative chemotherapy may be required. Incisional biopsy of the tumor, peritoneal deposits, and excised specimen. There was no history of weight loss or anorexia. There was no history of any chronic illness/comorbidities. Her personal, psychosocial, and family histories were unremarkable. On examination, her vitals were stable, and she had pallor. Her abdominal examination revealed tenderness over the left iliac fossa, hypogastric, and umbilical regions. Per rectal examination revealed stool loaded with increased sphincter tone. The rest of the systemic reviews were unremarkable.Her laboratory investigations showed anemia, leucocytosis, and stool for occult blood was positive Table . An erecTo manage acute obstipation, a team consisting of a surgical oncologist, general surgeons, and a medical oncologist decided to go with an emergency exploratory laparotomy, which revealed a relatively mobile growth at the hepatic flexure. Deposits of size 2.5 × 2 cm Figure  were see2 given intravenously for 21 days, and a tablet capecitabine (antineoplastic) 850 mg/m2 per oral was given for 14 days. Each cycle was of 21 days. A total of 6 cycles of chemotherapy were given. The patient was followed up till 6 months post‐intervention, where no significant complications were detected. A follow‐up CT revealed no new developments at the last visit.The final histopathology reports Figure  showed n3In the pediatric age group, CRC is rare and affects approximately one per million population in the USA.Diagnosis can be made based on radiological and histological evaluation.Poles et al. found that age less than 21 years was significantly associated with mortality in CRC.4Early diagnosis and intervention in CRC are crucial for better outcomes. CRC should be suspected in pediatric patients with unspecified abdominal pain. The initial evaluation should include an erect X‐ray abdomen and a colonoscopy. If CRC is suspected, a biopsy during colonoscopy should be done. Additionally, a computed tomography scan of the chest, abdomen, and pelvis will help diagnose, stage, and plan intervention. Before planning for surgery, a baseline CEA level should be obtained. Surgical intervention in advanced disease mainly includes proximal diversion, bypass, and bowel resection with anastomosis. Following surgery, postoperative chemotherapy may be required.Chintapenta Harshini: Conceptualization; data curation; investigation; project administration; writing – original draft; writing – review and editing. T Sri Harsha: Conceptualization; data curation; investigation; project administration; writing – original draft; writing – review and editing. Billakanti Rajkumar: Conceptualization; data curation; investigation; project administration; supervision; writing – original draft; writing – review and editing. Humaira Shaikh: Conceptualization; data curation; investigation; project administration; writing – original draft; writing – review and editing. Nooreen Habib: Data curation; investigation; writing – original draft; writing – review and editing. Ayush Anand: Conceptualization; investigation; project administration; supervision; validation; visualization; writing – original draft; writing – review and editing.The authors did not receive any funding for this article.The authors have no conflict of interest to declare.A written informed consent was obtained from the patient's legal guardian based on the journal's policies.


Giant prolactinoma (> 4 cm in dimension) is a rare disorder. Invasive macroprolactinoma has the potential to cause base of skull erosion and extend into the nasal cavity or even the sphenoid sinus. Nasal bleeding caused by intranasal tumor extension is a rare complication associated with invasive giant prolactinoma.We report a case of giant invasive macroprolactinoma with repeated nasal bleeding as the initial symptom.A 24-year-old man with an invasive giant prolactinoma in the nasal cavity and sellar region who presented with nasal bleeding as the initial symptom, misdiagnosed as olfactory neuroblastoma. However, markedly elevated serum prolactin levels (4700 ng/mL), and a 7.8-cm invasive sellar mass confirmed the diagnosis of invasive giant prolactinoma. He was treated with oral bromocriptine. Serum prolactin was reduced to near normal after 6 months of treatment. Follow-up magnetic resonance imaging showed that the sellar lesion had disappeared completely and the skull base lesions were reduced.This case is notable in demonstrating the aggressive nature of untreated invasive giant prolactinomas which can cause a diagnostic difficulty with potential serious consequences. Early detection of hormonal levels can avoid unnecessary nasal biopsy. Early identification of pituitary adenoma with nasal bleeding as the first symptom is particularly important.The online version contains supplementary material available at 10.1186/s12902-023-01345-y. Invasive giant prolactinoma is one of the rare subtype of prolactin (PRL)-secreting pituitary neuroendocrine tumors (Pit-NETs), measuring > 40 mm, accounting for only 0.5% of all Pit-NETs, and 4.4% of all prolactinomas, often presenting with headaches, vision loss, hyperprolactinemia, hypopituitarism as the first clinical manifestations –3. TheraA 24-year-old young man presented initially with nasal bleeding as the first symptom. He was admitted to West China Hospital of Sichuan University due to “intermittent nasal bleeding for 2 years and vision loss for 6 months”.The patient had a 2-year history of infrequent nasal bleeding and intermittent episodes of nasal bleeding. He had reported no nasal obstruction or dizziness, and nasopharyngoscopy in other hospitals had revealed no abnormalities. He had no nausea, anorexia, weakness, weight gain, gynecomastia, fatigue, or other symptoms, and he was taking no medication. However, 6 months before presentation at our hospital, following repeated epistaxis the patient developed blurred vision in both eyes, and his visual field decreased and gradually worsened. He reported intermittent mild headaches but no sinus pain, numbness, tingling, or constitutional symptoms. The patient did not report sexual dysfunction, including decreased libido or erectile dysfunction. His family history was unremarkable and he had no history of traumatic injury or brain surgery before admission.2. He did not have acromegalic or cushingoid features. The remainder of the examination was normal.On physical examination, his temperature was 36.5 °C, his pulse was 83 beats per minute, his blood pressure was 119/75 mmHg, and his respiratory rate was 19 breaths per minute. His height was 169.0 cm, his weight was 67.7 kg, and body-mass index  was 23.7 kg/mNasopharyngeal magnetic resonance imaging (MRI) and nasopharyngeal biopsy were performed. Pituitary MRI showed an irregular, abnormal signal mass in the nasal cavity, sphenoid sinus, and sellar region, about 7.8 × 4.6 × 5.0 cm in size Fig. A, B, C. The patient was diagnosed with olfactory neuroblastoma and was scheduled to undergo surgery, supplemented by radiotherapy and chemotherapy. The main reason for the impaired vision and visual field was considered to be that the suprasellar lesions were compressing the optic chiasm. To save vision and the visual field, otolaryngologists planned to work with neurosurgeons to remove the upper lesions of the saddle in order to decompress the optic nerve, followed by radiotherapy of the nasopharynx. However, after carefully reading the MRI results, we determined that the lesion was invasive and mainly located in the sphenoid sinus and the cavernous sinus and saddle. There was no obvious space in the ethmoid sinus or nasal cavity, which is not completely consistent with the characteristics of olfactory neuroblastoma. Routine preoperative examination of pituitary and target-glands related hormones showed the initial PRL level of this patient was greater than 200 ng/mL, and the measured value after dilution was as high as 4700 ng/mL, with normal thyroid-stimulating hormone (TSH), free thyroxine (FT4), luteinizing hormone (LH), follicle-stimulating hormone (FSH), testosterone (T) and cortisol levels Table . CombineHe was administered oral bromocriptine at an initial dose of 2.5 mg, once a day because of difficulties in obtaining cabergoline in Chengdu. After 3 days, the dose was increased to 2.5 mg twice a day. No nausea, vomiting, or other discomfort followed. The oral bromocriptine was gradually increased after 1 week to 2.5 mg, 3 times per day. He has tolerated the bromocriptine well without any adverse events. The PRL levels were reviewed each month, and the MRI (plain scan + enhancement) was repeated at 6 months and 12 months after treatment. Prolactin levels had reached normal or near-normal level within approximately 6 months. After 12 months of follow-up, the patient’s visual acuity and visual field improved significantly, and PRL had gradually normalized. The nasopharynx was examined, and we found that the nasopharynx mucosa was chronically congested; the nasopharynx, the double pharyngeal crypt, the eustachian tube bulge, and the surface of the pharynx were smooth. Magnetic resonance imaging of the sellar region showed a significant reduction in the size of the giant macroprolactinoma.We present a case of a 24-year-old male patient diagnosed with a giant prolactinoma invading the nasal cavity. Rarely for an invasive giant prolactinoma, the initial presenting symptom was nasal bleeding. Prolactinoma is one of the most common pituitary adenomas among Pit-NETs, with a prevalence of 6–10 to 60 cases per 100,000 patients. Prolactinomas are categorized based on their size, with microadenomas measuring < 1 cm, macroadenomas measuring > 1 cm, and giant prolactinomas, which are characterized by size > 4 cm, and PRL levels > 1,000 ng/mL [Most giant prolactinomas are invasive, and can have suprasellar extension, invasion to cavernous and sphenoid sinuses and clivus and rarely posteriorly into the brainstem, but they do not have biological characteristics of malignant tumors. Patients with giant pituitary prolactinoma may have a higher incidence of visual disturbances . Other sThe patient had normal libido, no sarcopenia and testosterone levels were normal. The possible explanation is that this patient retains an intact pituitary–gonadal hormonal axis. There have been many reports of this phenomenon, such as Shimon et al., which reported that 40% of men presenting with prolactinomas presented with normal or borderline testosterone levels . Ono M ePituitary adenomas presenting with nasal bleeding as the first symptom are extremely rare. In 1989, Van der Mey reported three patients with pituitary adenoma invading the nasal cavity. Of those patients, two with pituitary adenoma first presented with epistaxis . BecauseIn the present case, our patient had repeated nasal discharge as the first symptom, and nasal laryngoscopy revealed a new organism in the right olfactory sulcus. Olfactory neuroblastoma was suspected. However, the significantly increased serum PRL(> 200 ng/ml) excluded the possibility. The initial PRL level of this patient was greater than 200 ng/mL, and the measured value after dilution was as high as 4700 ng/mL. The main reason for this phenomenon may be the hook effect, which should be considered in all cases of large prolactinomas with normal or mildly elevated PRL levels . The hooWhen a pituitary adenoma invades the nasal cavity, it needs to be differentiated from some primary nasal tumors, such as nasopharyngeal malignant lymphoma and olfactory neuroblastoma , althougThe goal of treatment for large invasive prolactinomas is to reduce tumor volume and serum PRL levels. In the case presented here, the MRI of the saddle area showed a large tumor; the pituitary PRL level or pituitary imaging had not been previously screened. The level of PRL found in this case was significantly higher than normal. At present, the first-line treatment for giant prolactinomas is medical with dopamine agonists . A meta-In the case reported here, since it was difficult for our patient to obtain cabergoline at that time, oral bromocriptine was preferred. The PRL was significantly reduced after 6 months by gradually increasing the dose. A follow-up MRI scan showed that the lesions in the sellar region had nearly totally vanished, and the lesions in the skull base and nasal cavity were reduced. Additionally, the patient’s visual acuity improved, and there was no more nasal hemorrhage.In summary, pituitary adenomas rarely invade the nasopharyngeal cavity, especially with repeated nasal bleeding as a first reported symptom. These pituitary adenomas presenting as nasopharyngeal masses can cause a diagnostic difficulty with potential serious consequences, such as adopt unnecessary treatments and delay optimal treatments, increase the potential side effects and economic burden. Therefore, we emphasize the importance of radiologic studies and hormone detection. Once the diagnosis of prolactinoma is established, dopamine agonist should be the first choice for treatment.Additional file 1.


Hemolymphangioma, a rare type of lymphangioma, is a benign hamartoma of the blood vessels and lymphatic system. Considered to be extremely rare among adults with infrequent occurrence in abdominal regions, lymphangioma involving mesentery accounts for <1 % of cases and only 0.05 % involve the GI tract. Due to the absence of typical clinical presentation, making a confirmatory preoperative diagnosis is challenging.An 18-year-old Bangladeshi man visited the hospital with the complaints of epigastric and umbilical pain for 15 days which was insidious in onset. Physical examination revealed an ill defined lump that was palpable on the right side of the abdomen. Ultrasonography showed multiloculated cystic mass having septation approximately 13 × 6 cm in size. An abdominal CT scan showed cystic lesion with septations measuring about 14.5 × 12.3 cm, compressing the bowel loops towards left, that was suggestive of mesenteric lymphangioma. The patient underwent partial surgical resection. The excised mass was sent for histopathology. Histopathology disclosed a benign tumor composed of proliferation of blood vessels of different sizes lined by endothelium in a fibromuscular stroma.Mesenteric lymphangiomas are infrequent malformations and very few cases of mesenteric hemolymphangioma have been reported in adults. Histopathology is required for confirmatory diagnosis and immunohistochemistry is required to differentiate the tumor subtype. Surgical resection is deemed a standard treatment modality.We report an extremely rare case of mesenteric hemolymphangioma to bring it to concern that even with the vague clinical presentations and involvement of age groups beyond the status quo, surgeons must be vigilant about abdominal MCL/hemolymphangioma and proceed accordingly. •Cystic lymphangioma rarely involves the abdominal regions and the majority of those involving mesentery become symptomatic before the age of 2.•We report an unusual case of surgically confirmed large hemolymphangioma in an 18-year-old Bangladeshi man.•Preoperative diagnosis and treatment plan relies on radiological imaging as establishing clinical suspicion is highly challenging due to the lack of typical presentations.•Since confirmatory diagnosis is reached through a postoperative histopathological examination and immunohistochemistry, financial limitations and lack of availability of high-end diagnostic techniques can act as a barrier to establish the same.•Laparotomy followed by partial surgical resection was carried out as a standard treatment modality.•A thorough follow-up following surgery is necessary to detect any signs of recurrence. Cystic lymphangiomas in adults make up 7 % of abdominal cysts . Of all In this, we report a rare case of surgically confirmed large hemolymphangioma in an adolescent male which presents its importance in highlighting the diagnostic limitations regarding the case in an economically disadvantaged society where accessibility to high-end diagnostic techniques even in a tertiary level hospital is not a given and hence, raising awareness to be vigilant regarding the workup in such a setting. It also puts forth a great message in the realm of surgical faculty that without prior experience of a rare case as such, the diagnostic suspicion is less likely to build as it was the first ever case of mesenteric hemolymphangioma that the surgical team dealt with in our medical college and hospital. This report has been written in line with the SCARE criteria .2An 18-year-old young man was admitted with the complaints of epigastric and umbilical pain for 15 days that was insidious in onset and dull aching in nature. The intensity of the pain was mild to moderate. The pain was not related to any specific aggravating factor including food intake. Pain was relieved by taking oral medications. He noticed a gradual increment in the intensity of pain for 7 days. He also complained of fever during the last 7 days that was continuous in nature. It was not associated with chills and rigor. The highest temperature recorded was 101 degree Fahrenheit that decreased with the intake of antipyretic. He did not recall vomiting and did not experience weight loss, cough, hematemesis, malena and jaundice. His bowel and bladder habit was normal. There were no major ailments in the past. He had no medical history of abdominal trauma or surgery and no family history of malignant tumor. He belongs to a poor socio- economic background and maintains satisfactory personal hygiene.2. He presented with an increased body temperature, anemia was not evident, he was non icteric, Virchow's gland and accessible lymph nodes were impalpable. On inspection, the abdomen was flat with centrally placed, inverted and vertically slitted umbilicus, and intact hernial orifices. There was no visible swelling and peristalsis. On palpation, the abdomen was mildly tender over the epigastrium, umbilicus, right hypochondriac and right lumbar region. An ill-defined intra abdominal lump was found involving the right hypochondriac and lumbar region which had a smooth surface, was soft in consistency, partly mobile from side to side but not above downwards, non-pulsatile and not moving with respiration. On auscultation, bowel sound was present which was normal. No bruit was audible. Digital rectal examination revealed no abnormalities. No other systemic abnormalities were observed.On physical examination, the patient was ill-looking and co-operative with a BMI of 19.92 kg/mAfter a thorough history-taking and examination, we proceeded for laboratory investigations, imaging, and histopathology as a part of diagnostic workup. No abnormality was detected in laboratory investigations including Complete Blood Count (CBC), Serum amylase, lipase, electrolytes, creatinine, and Random blood sugar (RBS). Echinococcal antibody was also negative. X-ray abdomen was found to be normal. Ultrasonography (USG) showed multiloculated complex cystic mass having septation approximately 13 × 6 cm in size on the right side of the abdomen. We then carried out ultrasonography guided fine needle aspiration cytology of the abdominal mass which showed smears with lymphocytes and foamy histiocytes in a background of thick proteinaceous material. No evidence of malignancy was seen and the findings were compatible with lymphangioma. Contrast enhanced Computed Tomography (CECT) scan was performed and the findings of CECT were consistent with cystic lesion with septations noted at right side of abdomen, measuring about 14.5 ∗ 12.3 cm, compressing the bowel loops towards left. After IV contrast it showed peripheral and septal enhancement. No evident solid component was detected. It showed no abnormality in liver, gallbladder, pancreas and kidneys and was suggestive of mesenteric lymphangioma. Origin of the lesion could not be mentioned .Fig. 1CoFollowing the challenging diagnostic workup, with the patient's informed written consent we concluded the surgical approach to be our definitive modality of treatment. We conducted a thorough preoperative assessment to prepare the patient for surgery. Thereafter, laparotomy followed by partial surgical resection of the mesenteric lymphangioma was carried out along with adhesiolysis and hemostasis. The margin at the base of the lesion could not be cleared. Patient had been placed under general anesthesia with endotracheal intubation in supine position a,b,c,d. Our patient had a smooth surgical recovery devoid of any complications and a follow-up after two months of surgery revealed no signs of recurrence .Fig. 3Cl3Mesenteric lymphangiomas are infrequent congenital malformations that manifest as a non-epithelial, neoplastic proliferation of lymphatic channels . The defHemolymphangioma, originating from the mesenchymal tissue, is a benign hamartoma of the blood vessels and lymphatic system . This isIn case of large abdominal hemolymphangioma, patients may present with acute abdominal pain and discomfort, and bowel obstruction . ClinicaIn particular, specific enhanced characteristics can be observed on imaging with regard to the differing size of blood vessels in hemolymphangioma . In tumoAccording to the imaging findings, we presumed it to be a case of mesenteric cystic lymphangioma but later postoperative histopathological examination showed hemorrhagic components compatible with a hemangioma. Since the radiological findings in our case was not typical of a hemangioma, the discrepancy between the imaging findings suggestive of a lymphangioma and surgical pathology reports compatible with hemangioma could have been eliminated through immunohistochemistry (IHC). In contrast, the poor socioeconomic background of the patients can act as a hindrance in opting for expensive diagnostic evaluations in case of third world countries such as Bangladesh. Given the underprivileged social position of our patient and lack of availability of high-end diagnostic techniques in our medical college and hospital, we could not carry out an immunohistochemistry examination.Overall, it may be possible to achieve the best results with a reduced recurrence rate by complete excision; however, thorough follow up is required . There h4We report an extremely rare case of mesenteric hemolymphangioma to bring it to concern that even with vague clinical presentations and involvement of age groups other than the usual majority as evident in this case, surgeons must not completely exclude the suspicion of abdominal MCL/hemolymphangioma as a differential and should proceed accordingly.Similarly, we aim to highlight the importance of roles of imaging techniques in accurate preoperative diagnosis and surgical planning. Patients will be highly benefited if radiologists are more aware of the radiological findings of MCL/hemolymphangioma especially in an economically disadvantaged social setting where patients can't afford expensive diagnostic tests like immunohistochemistry.Having said that, the requirement of histopathology in making a confirmatory diagnosis and immunohistochemistry to differentiate the tumor subtype should also be given the utmost importance while managing such cases.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-chief of this journal on request.Not commissioned, externally peer-reviewed.Nothing to declare.The author(s) received no financial support for the research, authorship and/or publication of this article.Dr. Asmita Bhusal was involved in the ideation, data collection, drafting, designing and editing of the manuscript, and took entire responsibility for publishing this study under the supervision of Dr. Quazi Habibullah and Dr. Mashiur Rahman.Dr. Quazi Habibullah was the chief surgeon of the case.Dr. Mashiur Rahman and Dr. Biplob Bandh reviewed the manuscript.Tanvin Dola, Dr. Saiful Islam and Dr. Saugat Bista helped in writing the manuscript.All the authors read the paper and gave final approval of the version to be published.Dr. Asmita Bhusal and Dr. Quazi Habibullah accept full responsibility for the work and/or the conduct of the study, had access to the data, and controlled the decision to publish.Not applicable.None to declare.


In recent years, there has been a notable increase in acute diverticulitis cases, attributed to modern lifestyles and improved diagnostic techniques. We present a rare case of concurrent acute appendicitis and diverticulitis in a 35-year-old male who came to the emergency department with abdominal pain. While appendicitis typically requires surgery, diverticulitis is often managed conservatively. Computed tomography is key in diagnosis and decision-making. Despite their differing treatments, cases like this challenge the perception of their rarity.This case prompts consideration of multifocal abdominal pathology. Recognizing concurrent appendicitis and diverticulitis is crucial for guiding appropriate diagnostic and treatment strategies, potentially including non-operative management in select cases. Appendicitis and diverticulitis stand as two of the most prevalent etiologies for acute abdomen frequently encountered in the emergency room setting. While these conditions individually contribute significantly to the burden of acute abdominal pathology, the occurrence of both simultaneously is considered exceedingly rare. Appendicitis requires a surgical approach for resolution, in contrast with the conservative management typically employed for uncomplicated diverticulitis ,2.Acute appendicitis, characterized by inflammation of the vermiform appendix, is a well-recognized surgical emergency. Prompt surgical intervention remains the cornerstone of treatment, resulting in favorable outcomes when executed expeditiously . ConversGiven their disparate management paradigms, the co-occurrence of appendicitis and diverticulitis presents a diagnostic and therapeutic challenge. Limited literature exists addressing this unique clinical scenario, emphasizing the rarity of such cases. Therefore, we present a case of concurrent acute appendicitis and uncomplicated diverticulitis, highlighting the clinical presentation, diagnostic approach, and successful surgical intervention. A 35-year-old male, with a history of radical right orchiectomy for seminoma, presented to the emergency department with a 24-hour history of abdominal pain localized in the right and left iliac fossas. The pain had progressively worsened in the last few hours, accompanied by an increase in bowel movement frequency. He denied experiencing any other associated symptoms.On examination, tenderness was noted in the lower abdomen, along with positive appendicular signs , and rebound tenderness was elicited on palpation of the left iliac fossa.3, N: 4,587-10,060/mm3) with a mild neutrophil predominance . There was a marked inflammatory response, as indicated by a C-reactive protein level of 100.1 mg/dL (N: <1.0 mg/dL).Blood analyses revealed leukocytosis  of the abdomen and pelvis Figure  demonstrIntravenous fluids were initiated, and the patient was started on antibiotics (ampicillin + sulbactam 3 g IV) and provided with analgesia. A diagnostic laparoscopy was proposed.The surgical exploration unveiled an inflamed appendix, as depicted in Figure The patient's postoperative course was uneventful. Liquid oral intake was reintroduced six hours after the surgery and was well-tolerated. The patient was discharged on postoperative day 3, with oral antibiotics prescribed for an additional five days.The histopathology report confirmed a suppurative acute appendicitis with fibrous obliteration of the tip, along with evidence of acute serositis.The coexistence of acute appendicitis and uncomplicated diverticulitis in the same patient is a rare clinical entity, often posing a diagnostic challenge. We present a case that exemplifies this unusual occurrence, prompting a comprehensive discussion on its clinical implications and management strategies.To the best of our knowledge, only a limited number of cases with concurrent appendicitis and diverticulitis have been reported in the literature. Notably, a case described by Hussain et al. in 2016 shares similarities with our present case . In theiFurthermore, in a review of the literature, we identified another case of concurrent appendicitis and diverticulitis reported by Shademan et al. in 2013, as depicted in an image of a CT in their article . SimilarWhile surgical intervention remains the cornerstone of treatment for acute appendicitis, there is a growing body of evidence supporting the non-operative management of uncomplicated cases with antibiotics . This apAccording to Turner et al., there has been a noted rise in the incidence of acute diverticulitis in recent years, potentially attributed to modern lifestyle factors and enhanced diagnostic capabilities, particularly the increased judicious use of imaging techniques . It is cThe coexistence of acute appendicitis and diverticulitis, though traditionally deemed rare, may be more prevalent than initially perceived. This case underscores the importance of thorough diagnostic evaluation when confronted with atypical presentations of abdominal pain. It challenges the conventional approach of immediate surgical intervention in young male patients with suspected appendicular disease, advocating for a more comprehensive evaluation, including computed tomography.This case serves as a noteworthy contribution to the existing body of literature, providing clinicians with valuable insights into the management of these uncommon dual pathologies. Additionally, it underscores the significance of considering multifocal abdominal pathology when confronted with atypical clinical presentations. The successful surgical management in this case aligns with the standard of care for acute appendicitis. However, the growing body of evidence supporting non-operative management for uncomplicated cases raises intriguing prospects for future exploration, especially in cases with concurrent pathologies.


The myxofibroma (MF) constitutes an uncommon, non-malignant, odontogenic neoplasm with potential mesenchymal derivation. The occurrence rate of this particular tumor is estimated to be around 0.05 new cases per million individuals annually. MFs exhibit a higher incidence rate within the age range of 10 to 30 years. The prevalence of these tumors is higher among the female population, with a predominant localization in the mandible, specifically in the posterior region. A female patient, 66 years old, was referred to the Department of Oral Surgery, Surgical Implantology and Radiology, Thessaloniki, Greece, complaining of a tumorous lesion in the anterior area of the maxilla and mild pain. Clinically, a solid in palpation lobulated tumor, covered by normal coloured mucosa was observed at the left upper incisor. After the excisional biopsy, the microscopic appearance of abundant fibromyxoid stroma, in particular, myxoid stroma intermingled with collagenous tissue, covered by stratified squamous epithelium, suggested the diagnosis of peripheral myxofibroma. During a 2-year follow-up, no recurrence was referred. This case illustrates the necessity of proper differential diagnosis of every tumorous lesion of the gingiva and of using the histopathological examination. The myxofibroma (MF) constitutes an uncommon, non-malignant, neoplasm with potential mesenchymal derivation that was initially documented by Virchow in 1863 . MFs areA female patient, 66 years old, came to the Department of Oral Surgery, Surgical Implantology and Radiology, School of Dentistry, Aristotle University of Thessaloniki, Greece, complaining of a tumorous lesion in the anterior area of the maxilla and mild pain due to food impaction in the interdental area. Before the examination, the patient provided written informed consent. This form was approved by the School of Dentistry, Aristotle University of Thessaloniki, and was in accordance with the Helsinki Declaration for research and patient ethics. Subsequently, the patient was examined thoroughly and referred for further examination to the Department of Oral Medicine and Pathology, School of Dentistry, Aristotle University of Thessaloniki, Greece. Clinically, a sessile, lobulated movable solid in palpation tumor was observed, covered by normal to erythematous mucosa, which derived from the gingiva of #21. The differential diagnosis included common entities such as fibroma, peripheral ossifying fibroma, giant cell tumor, and less common entities such as myxoma, ameloblastoma, ameloblastic fibroma, odontogenic fibroma or odontogenic keratocystic tumor. Figure The surgical procedure took place in the Department of Dentoalveolar Surgery, Surgical Implantology and Radiology and was carried through in a conservative fashion. A scalpel #15 was used for the excision and precautions were taken, to avoid any unnecessary removal of soft tissue, which could undoubtedly lead to gingival recession and further aesthetic complications for the patient. Subsequently, sutures were placed, and the surgical area was covered with surgical cement. The stepwise surgical procedure is displayed in Figure The tissue specimen was preserved in formaldehyde and submitted to histopathological examination in the Department of Oral Medicine/Pathology of the same Institution. Microscopically, an abundant fibromyxoid stroma, covered by stratified squamous epithelium, which presents hyperplasia and spongiosis was seen. The myxoid stroma was intermingled with collagenous tissue with numerous inflammatory cells. Scattered spindle-shaped cells, within the myxoid stroma, admixed with lymphocytes and plasma cells were also noticed. Nuclear atypia or mitoses were not identified. Therefore, the diagnosis of peripheral MF was established Figure .The recall of the patient took place two weeks later where the successful wound closure was observed and only an erythematous area was still present, due to the neovascularization process Figure .During a 2-year follow-up, no recurrence was referred.Central type of MFs possess a tendency for expansion, primarily manifest in the jaws, with a significantly high predilection for this anatomical region. The mandible exhibits a higher incidence of involvement compared to the maxilla -15, withThe majority of MFs exhibit no symptoms, although a small number of patients have reported experiencing escalating pain due to the invasion of adjacent structures . EvidentRegarding the remaining odontogenic tumors, the conclusive diagnosis of MFs relies on the assessment of histopathological characteristics. If the size of the lesions is substantial, it may be necessary to conduct a biopsy in order to determine the characteristics of the tumor and develop an appropriate therapeutic strategy. The preferred treatment modality, as reported in the literature, is surgical excision . There iThe initial differential diagnosis included common entities such as fibroma, peripheral ossifying fibroma, giant cell tumor, and less common entities such as myxoma, ameloblastoma, ameloblastic fibroma, odontogenic fibroma or odontogenic keratocystic tumor. The best case scenario would be the fibroma, but since 100% accuracy and certainty are not possible based solely on clinical features, any tissues, after removal, should be submitted to histopathological examination, regardless of how benign they appear to be. The peripheral location of the MF allowed for a conservative excision, avoiding any further functional or aesthetic complications. During a 2-year follow-up, no recurrence was referred.


Periarticular chondromas are common in the humerus and femur but rarely occur in the temporomandibular joint. We report a case of a chondroma in the anterior part of the ear. One year prior to his visit, a 53-year-old man became aware of swelling in the right cheek region which gradually increased in size. In the anterior part of the right ear, there was a palpable 25 mm tumor, elastic and hard, with poor mobility and without tenderness. A contrast-enhanced computed tomography CT showed a mass lesion with diffuse calcification or ossification in the upper pole of the parotid gland and areas of poor contrast within. A magnetic resonance imaging showed a low-signal mass lesion at the parotid gland with some high signals in both T1 and T2. Fine-needle aspiration cytology did not lead to diagnosis. Using a nerve monitoring system, the tumor was resected with normal tissue of the upper pole of the parotid gland in the same way as for a benign parotid tumor. Distinguishing between pleomorphic adenoma, including diffuse microcalcification of the parotid gland and cartilaginous tumors of the temporomandibular joint, may be sometimes difficult. In such cases, surgical resection may be a beneficial treatment option. Chondromas  are benign tumors of a mesenchymal origin that consist of mature hyaline cartilage nodules. Depending on their origin, they are classified into three clinical types. Enchondromas make up 80% of the total cases. They occur in the medulla and represent variants of condylar hyperplasia and metaphysis . Juxta-aThe temporomandibular joint (TMJ) is a rare site of occurrence for chondromas. Conversely, the preauricular region is the most common site for parotid gland tumors, such as pleomorphic adenoma, Warthin’s tumor, or a parotid carcinoma, but not a chondroma. Furthermore, patients with a chondroma of the TMJ sometimes complain of clicking, pain of the TMJ, or difficulties in opening their mouth due to temporomandibular disorders. Patients with a chondroma of the TMJ may therefore initially be diagnosed with parotid gland tumors or inflammation of the TMJ. Herein, we report a rare case of juxta-articular chondroma that was in contact with the TMJ and partially extended to the upper pole of the parotid gland. The current case presents a difficult differential diagnosis of periauricular tumors between a benign tumor of the upper pole of the parotid gland and cartilaginous tumors derived from the TMJ and a surgical approach based on a procedure for a parotid benign tumor.A 53-year-old man presented an elastically hard, poorly mobilizable tumor (25 × 25 mm) in the right anterior auricular region. The tumor had gradually enlarged over the course of a year. There was no evidence of facial nerve palsy, clicking, pain, or mouth-opening difficulties due to temporomandibular disorders.18F-fluorodeoxyglucose (18F-FDG-PET) showed a mild uptake of 18F-FDG (SUVmax = 3.25) (Computed tomography (CT) showed an oval tumor in the region of the right TMJ, extending to the upper pole of the parotid gland. The tumor was oval-shaped with an actual width of 2.5 cm and a length of 3 cm. It had well-defined borders and a diffuse hyper-absorptive zone within, which indicated diffuse calcification A,B. Posi = 3.25) C. There  = 3.25) D, while  = 3.25) E. The co = 3.25) F. Fine-nConsidering the possibility that the temporal branch of the facial nerve might have run near the tumor, we performed the same surgical procedures as those that would be used for a parotid gland tumor. Briefly, after a preauricular S-shaped skin incision, the capsule of the superficial parotid gland was exposed. Using a facial nerve monitoring system, NIM-NEURO™3.0 , the main trunk of the facial nerve was confirmed near the stylomastoid foramen. The temporal branch of the facial nerve was exposed in a peripheral direction. The tumor was located at the upper pole of the parotid gland and was in contact with the TMJ capsule and the temporal branch of the facial nerve. Meanwhile, direct contact with the external auditory canal was unclear, and there was no obvious adhesion to the surrounding parotid tissue or facial nerve A. The tuMicroscopically, a well-circumscribed lobulated tumor was found within the parotid gland. The cellularity in the lesion was low, and isolated basophilic cells with indistinct cytoplasmic borders were scattered in the eosinophilic to basophilic chondroid matrix. Calcified materials were diffusely deposited in the tumor. Lymphocytic infiltration with foreign-body giant cells and a few Touton giant cells were distributed mainly along the interlobular septa and in the periphery of the tumor A. DirectAfter surgery, the patient developed mild palsy of the temporal branch of the facial nerve and was treated with oral prednisolone with a dose tapered from 30 mg for one week. The cause of the paralysis was thought to be nerve extension due to the detachment procedure during the surgery. This facial palsy was completely healed two weeks after the surgery. The patient was followed with an ultrasound examination every three months for the first year and then every six months for two years. A plain MRI examination at six months and two years after the surgery confirmed no evidence of recurrence. At the time of the final checkup, both oral and written consent were obtained from the patient, and the Wakayama Medical University Institutional Review Board approved publication of this case report .Chondromas usually occur proximal to articular capsules and tendons and rarely occur outside of the skeleton or in soft tissues. These tumors are thus rarely found in the TMJ and preauricular region ,9. ChondPleomorphic adenomas, the most common type of benign parotid tumor, may contain chondrogenic differentiation. Owing to the difficulty in distinguishing from cartilage by imaging, strict morphologic criteria are necessary to rule out benign adenomas. Chondromatous degeneration via the accumulation of mucoid material around the myoepithelial cells in pleomorphic adenomas of parotid glands may resemble a chondroma. Coelho et al. described the characteristics of the imaging of pleomorphic adenoma with calcification as follows: “(i) a smoothly marginated mass that is hypoechoic on ultrasound, (ii) a higher degree of attenuation in the tumor than in the surrounding parenchyma on CT, and (iii) low T1-weighted and high T2-weighted signal intensity on magnetic resonance imaging (MRI)” . If the 18F-FDG-PET is reported to be a useful examination for evaluating the potential or grade of the malignancy of chondroid tumors [Chondrosarcoma is also a relatively rare type of tumor, but it should be ruled out for the differential diagnosis of malignant diseases around the TMJ. In general, chondrosarcoma pathologically shows increased cellularity with atypical findings, such as binucleated chondrocytes. In the current case, some atypical cells with unclear cell boundaries were observed. Some cases of chondrosarcoma were thought to arise via the malignant transformation of chondroma . Recentld tumors . SUVmax The pathogenesis of chondromas is thought to be osteochondromal exostosis or the proliferation of germ cells with chondrogenic potential due to abnormalities in the direction of osteochondral growth. Calcification occurs as a result of development or regression of the lesion over a long period of time ,17. HistHistologically, most extraskeletal chondromas have a mature hyaline cartilaginous appearance, and approximately one-third exhibit focal or diffuse calcification. In our patient, diffuse prominent calcification and fibrohistiocytic reaction obscured the chondroid appearance . These findings made this case mimic tumoral calcinosis and made the diagnosis difficult. It is important to detect cartilage with careful observation for the differentiation between tumoral calcinosis and an extraskeletal chondroma. Immunohistochemically, chondrocytes are positive for S100, while epithelial and myoepithelial cells, which are not typically present in chondromas, are negative for S100. In the current case, there were cells that were weakly positive for S100, but there were no epithelial or basal cells that were positive for CK and p63. In addition, the von Kossa stain was positive; therefore, the current patient was diagnosed as having a chondroma with prominent calcification. Moreover, this case was considered a juxta-articular or periosteal chondroma in close proximity to the TMJ because it was located posterolaterally to the mandibular condyle and had no contact with the articular surface.WHO Classification of Tumours (5th Edition), Soft Tissue and Bone Tumours, 2020, describes findings on the cytology of cartilagenous tumors [Fine-needle aspiration cytology is a useful and less-invasive examination for the preoperative diagnosis for head and neck neoplasms, even for cartilaginous tumors. The As a policy for surgery, we decided to treat using the same strategy for benign tumors of the parotid gland because of the relatively high recurrence rate of chondromas, including other sites in previous reports. Additionally, some pleomorphic adenomas are accompanied by calcification within the tumors ,21, whicThis case demonstrates the difficulty in differential diagnosis between a pleomorphic adenoma with chondrogenic differentiation of the upper pole of the parotid gland and a chondroid tumor of the TMJ. Differentiation via imaging findings is sometimes difficult, and diagnosis requires immunological findings with pathology if it is difficult to obtain enough cells by fine-needle aspiration.Surgical resection is recommended in such cases because of the possibility of malignant transformation and the relatively high recurrence rate, including occurrence in other sites, even in the case of benign chondroid species. Intra-operative nerve monitoring is helpful to preserve temporal branch of the facial nerve if the tumor is located inside the upper pole of parotid gland.


Nipple adenoma is a very uncommon, benign neoplasm that involves the nipple. A palpable mass of the nipple associated with nipple discharge and erosion or ulceration is the common clinical presentation. Generally, complete surgical excision of the nipple is the main treatment, alternative therapeutic methods such as Mohs micrographic surgery, nipple splitting enucleation, and cryotherapy can be considered. Disorders of the breast in young women are generally benign. Even if the management during pregnancy is usually conservative and surgical excision is reserved for very strong malignancy suspicion, benign lesions can cause the impossibility to breastfeed after giving birth when involving the nipple.We present the case of a 28-year-old female, who was referred to the Breast Unit of the University Hospital of Modena  in May 2020 with a 12-months history of enlargement of the left nipple with associated erythema, serohemorrhagic discharge, and pain in the left nipple region. The diagnostic assessment came out in favor of a nipple adenoma. After surgical treatment was recommended, the patient got pregnant. Taking into account the major risks of surgery during pregnancy, a multidisciplinary discussion was conducted, to consider whether to proceed with surgery or postpone it after pregnancy. Because of the volume and the position of the adenoma, the indication for surgical excision was confirmed, to allow regular lactation and breastfeeding immediately after giving birth and to avoid potential obstructive complications. Surgical excision of nipple adenoma without complete resection of the nipple was performed after her first trimester of pregnancy under local anesthesia. A histopathological examination confirmed the diagnosis. No recurrence occurred after 12 months. The patient gave birth, had no deficit in lactation, and successfully breastfed.Therefore, we consider that nipple adenoma enucleation might be a safe treatment even during pregnancy. Moreover, conservative local treatment of nipple adenomas can preserve the nipple aesthetically and functionally, thus allowing regular lactation and breastfeeding in young women.The online version contains supplementary material available at 10.1186/s13006-023-00554-4. Nipple adenoma is a rare benign breast proliferative process of lactiferous ducts that usually affects middle-aged women. A palpable mass of the nipple associated with nipple discharge and erosion or ulceration is the common clinical presentation . Nipple Disorders of the breast in young women are generally benign. Although rare, nipple adenoma and other benign breast disorders (BBD) can also be seen during pregnancy and lactation. Management of BBD during pregnancy is usually conservative and surgical excision is only mandatory in case of rapid enlarging or discordance in the triple diagnostic assessment .We report herein a case of adenoma of the nipple in a young woman, surgically managed during pregnancy by curative tumor resection without excision of the nipple. At last, we summarized the indications and management of nipple adenomas in pregnant women.We present the case of a 28-year-old female, who was referred to the Breast Unit of the University Hospital of Modena  in May 2020 with a 12-months history of enlargement of the left nipple with associated erythema, serohemorrhagic discharge, and pain in the left nipple region. Her past medical history was unremarkable. Positive family history of early-stage breast cancer was found in her mother, at the age of forty-eight. The patient had noticed the mass about 12 months before her consultation and during that interval the lesion had slowly increased in size, causing mild symptoms of pressure and discomfort.On physical examination, the left nipple was enlarged and twice the size of the contralateral, a non-tender well circumscribed movable mass of approximately 10 mm was palpable, as shown in Fig. On the fine-needle aspiration cytology (FNAC) examination serous-haematic material with low to moderate cellularity within epithelial hyperplastic cellular nests and myoepithelial units was reported. This pattern was suggestive of a benign adenomatous process  with no signs of atypia or malignancy . Fine-neThe surgical indication was to perform the excision of the mass under local anesthesia, with preservation of the nipple. During the waiting period for the surgical procedure, the patient became pregnant. She has been referred once again to our Breast Unit for scheduled mammary ultrasonographic control in her sixth week of pregnancy. Taking into account the major risks of surgery during pregnancy, a multidisciplinary discussion was conducted, to consider whether to proceed with surgery or postpone it after pregnancy. Because of the volume and the position of the adenoma, the indication for surgical excision was confirmed, to allow regular lactation and breastfeeding immediately after giving birth and to avoid potential obstructive complications such as galactocele, breast abscess, or mastitis. The end of the first trimester was established as the ideal time for the procedure. The patient underwent surgery in her fifteenth week of pregnancy. The surgical excision of nipple adenoma was performed under local anesthesia with mepivacaine by an experienced breast surgeon. A nipple splitting enucleation technique was performed through a 1 cm trans-nipple longitudinal incision made down through the long axis of the nipple profile, to expose and radically enucleate the mass, as shown in Fig. The postoperative course was uneventful. Outpatient monitoring showed a correct wound-healing process and a progressive reduction of the left nipple size. Figure Nipple Adenoma (NA), also known as erosive adenomatosis or florid papillomatosis of the nipple, is a benign proliferation of the lactiferous ducts of the nipple , 10. It This rare entity, which accounts for just 1–1.7% of benign breast lesions , can incDespite its benign behavior, the expanding growth pattern and frequent local recurrence could harm a patients’ quality of life, especially for women of childbearing age who need to maintain intact the function of the nipples. Moreover, the presence of an abnormal mass of the nipple could affect the baby’s ability to latch and suck during breastfeeding.Association with malignant breast carcinoma has been previously described; with regards to the probability of a tumor developing from nipple adenoma, no reliable data are available so far , 19.The optimal management of benign nipple lesions during pregnancy is controversial and scarce evidence has been produced . To our Regularly scheduled follow-up is recommended in these patients, regardless of the therapeutic methods, since nipple adenoma’s risk of recurrence or progressing into a malignancy can not be fully excluded.In conclusion, we reported a rare case of nipple adenoma and local excision in a pregnant young female. Traditional complete removal of the nipple may result in overtreatment, make breastfeeding impossible and an unsatisfactory aesthetic outcome. Conservative local treatment can be considered after the end of the first trimester of pregnancy and can allow the functional preservation of the nipple in young women.Additional file 1. Surgical Procedure – part1. Enucleation of left nipple adenoma with preservation of lactiferous ducts.Additional file 2. Surgical Procedure – part 2. Complete excision of left nipple adenoma.


Surgery is the only treatment for parastomal hernia (PH). When possible, stoma closure is the best way to manage this type of hernia, however, whether to perform it in a single approach with abdominal wall reconstruction (AWR) is still debatable. A 58-year-old woman with a type IV PH with loss of domain was submitted to preoperative optimization [botulinum toxin type A and progressive pneumoperitoneum (PPP)], followed by simultaneous stoma closure and AWR. Hospital discharge was on the eighth day with no complications. Six months later, no clinical evidence of recurrence or other complication was observed. Large PHs are technically challenging. Stoma closure and simultaneous AWR increase surgical risk. Preoperative optimization with a combination of adjuvants (including PPP) is feasible in PH and may overcome technical complexity, even though patient selection remains the key when choosing a PH repair with synchronous stoma closure. Parastomal hernia (PH) is the most common complication after stoma creation, tending to appear in the first 2 years. Incidence varies according to follow-up time, type of stoma, and diagnostic method, ranging from 50% to 78% in most series . Up to 7The only treatment remains surgery, despite lack of evidence for the best surgical option. Also, high morbidity and recurrence rates keep limiting surgical indication to only cases with disabling symptoms , 2, 5, 6Complexity increases when loss of domain (LOD) is present , since High recurrence rates after PH correction are frequently observed even when optimal treatment is offered . TherefoThe authors report a single-stage approach of a PH repair with a colostomy closure after preoperative BTA and PPP adjuvant techniques.2) woman with a history of arterial hypertension and hypothyroidism was submitted to Hartmann operation for acute diverticulitis Hinchey IV 2 years prior, complicated with superficial surgical site infection and wound dehiscence. PH was clinically evident 3 months after the index surgery.A 58-year-old overweight (BMI = 27 kg/mComputed tomography (CT) scan revealed a type IV PH (according to EHS classification ) with LOPatient was proposed to preoperative optimization with respiratory kinesiotherapy, adjuvant techniques (BTA and PPP), and one-stage surgery with stoma closure and hernia repair with a transverse abdominal release (TAR).et al. [BTA was applied 4 weeks before surgery according to our institution’s protocol: 500 U of Dysport® diluted in 20 ml of 0.9% saline distributed over 3 points on each side of the lateral abdominal wall between the costal margin and the iliac crest lateral to the semilunar line. Ten days before surgery the patient was admitted for PPP. An 8 Fr pigtail was inserted by interventional radiology, and a daily insufflation of 1 l of air was performed until a total volume of 7 l was reached , limitedet al. .One day before surgery, a comparative CT scan demonstrated increased abdominal cavity capacitance with bowel content totally reduced .et al. [Stoma closure with a colorectal anastomosis and PH type IV repair (midline and PHs) with a unilateral TAR (left side) were performed by placing a macroporous lightweight mesh in the retromuscular space, reaching the semilunar line on the right side. Antibiotic prophylaxis was done with 2 g cefazolin. Three drains were placed: one intraperitoneal, near the anastomosis; and two suctions drains at the retromuscular and subcutaneous planes. Intra-abdominal pressure and ventilation plateau pressure variation before incision and after closure were both close to zero. The procedure lasted 3 h and 44 min and was uneventful. The patient was admitted to the ICU, as protocolled in our institution, for total relaxation in the first 12 h and close monitoring, although no quaternary abdominal syndrome was expected according to predictive factors described by Quintela et al. .Empirical antibiotics were started on the third postoperative day due to fever and coagulation disorders, though no objective cause was detected. CT scan was performed excluding surgical complications. The patient was discharged from ICU to the nursery on the third day and from the hospital on the eighth day. Drains were removed on the fifth day. Patient was recommended to use abdominal binder for the first postoperative month. Six months later, no clinical evidence of recurrence or other complication were observed.PHs remain the Achilles tendon of the colorectal surgeon. Large PHs with LOD, associated with midline hernias, are technically challenging, with great abdominal wall disruption and myofascial retraction. Preoperative optimization with a combination of BTA and PPP, promoting abdominal volume expansion, is crucial to avoid serious postoperative complications such as abdominal compartment syndrome and hernia recurrence. Satisfactory results were described in a group of 16 patients with PHs who received the combination of BTA and PPP preoperatively, treated with the laparoscopic Sugarbaker technique .Although novel techniques are evolving for PHs, such as Pauli’s operation , which aTo our knowledge there are no previous reports of synchronous stoma closure and type IV PH repair after a preoperative combination of BTA and PPP.The adjuvants used in preoperative optimization minimize the need of more disruptive techniques. A step-up approach in abdominal wall reconstruction can be offered until myofascial closure and balanced intrabdominal and ventilation pressures are achieved. This leads to less physiologic derangement, which in turn enables one-stage procedure to be done with lower overall risk, even though patient selection remains the key when choosing a PH repair with synchronous stoma closure.


Up to 40% of Cushing’s disease (CD) patients show no evidence of an adenoma on dynamic contrast-enhanced MRI. Inferior petrosal sinus sampling (IPSS) remains the gold standard for diagnosis in these patients. Remission rates in MRI–Negative CD are far less at 50%–71%, compared with patients in whom an adenoma is identified on MRI. Endoscopic endonasal transsphenoidal surgery is the surgical approach of choice in these cases. Various adjuncts can be used to localize an adenoma. In this video, the authors highlight their additional usage of pituitary perfusion MRI for identification of the adenoma. They present their stepwise management algorithm and surgical techniques for sellar and suprasellar exploration in 6 cases of MRI–Negative CD operated on by the senior author (A.S.).https://stream.cadmore.media/r10.3171/2023.4.FOCVID2318The video can be found here:  The surgical approach of choice in these patients remains endoscopic endonasal transsphenoidal surgery.4 Various adjuncts can be used to localize an adenoma. In this video, we highlight our additional usage of pituitary perfusion MRI10 for identification of the adenoma. It utilizes the differential enhancement pattern of the normal pituitary gland, which is more avidly contrast-enhancing than the adenoma due to the differences in the vasculature of the normal and the adenomatous pituitary tissue. Pituitary perfusion curves thus help us identify the small adenomas which might be missed on routine dynamic MRI of the sella.In this video, we describe the management protocol and surgical techniques of exploration in 6 cases of MRI–Negative Cushing’s disease operated at our institute. Up to 40% of Cushing’s disease patients show no evidence of an adenoma on dynamic contrast-enhanced MRI.Our first patient is a 13-year-old female, a case of primary MRI–Negative Cushing’s disease with central gradient on IPSS. There was subtle thickening of the stalk on dynamic contrast-enhanced MRI of the sella, but there was no definite evidence of an adenoma. She underwent endoscopic endonasal transsphenoidal surgery with exploration of the pituitary gland. The patient positioning was as per the standard position used for an EETS, as shown here.A binostril approach was used. Due to the narrow surgical corridor in this pediatric patient, a middle turbinectomy was performed through the right nostril. A posterior septostomy was made. The sphenoid rostrum was drilled and nibbled and the sphenoid sinus was entered. Similar procedure was repeated through the left nostril. The sellar floor was drilled and eggshelled. Dura was opened from 5-o’clock to 7-o’clock position and the gland was explored. Suspicious adenomatous tissue was seen in the right side of the gland. It was curetted out and sent for histopathological analysis.Postoperatively, this patient had clinical and biochemical persistence of her disease, and histopathological examination did not reveal an adenoma. Thus, the patient underwent reexploration of the gland 2.5 months later.Repeat imaging revealed subtle thickening of the stalk without any evidence of an adenoma. Thus, a suprasellar exploration was performed via an extended endoscopic endonasal transsphenoidal approach. The tuberculum sellae and planum sphenoidale were drilled. There was no evidence of an adenoma on exploration of the gland. The dura over the planum was cut and the pituitary stalk was exposed. Adenoma was identified on the anterior pituitary stalk and was curetted out. Normal pituitary stalk and the chiasm were seen. Closure was performed using fascia lata, fat and augmented with fibrin glue, as there was intraop CSF leak.A lumbar drain was inserted in the immediate postoperative period as there was evidence of an intraop CSF leak. Nasal packs were removed after 48 hours. Lumbar drain was removed on postoperative day 5. There was no evidence of a CSF leak after the lumbar drain removal. The patient underwent clinical and biochemical remission, and histopathological examination revealed the presence of an adenoma.Our second case is a 12-year-old male with primary MRI–Negative Cushing’s disease. IPSS revealed a central gradient with right-sided lateralization. This patient underwent surgery through the standard endoscopic approach. There was conchal type of pneumatization seen in this patient, as evident on the preoperative CT. Intraoperatively, the identification of sella was difficult due to the conchal type of pneumatization. Moreover, the neuronavigation was malfunctioning, and thus we were not able to precisely identify and confirm the intraoperative anatomy. Extra bony drilling had to be performed to obtain adequate exposure. Bone was drilled to identify the sellar floor. Dura was identified and opened. What appeared to be the sellar floor actually was the planum dura in proximity to the tuberculum sellae, as confirmed after the dural opening and arachnoid bulge. Thus, a suprasellar exposure was not intended and was performed by mistake. Further drilling was performed inferiorly and the sellar floor could then be located. The sellar dura was opened. Adenomatous tissue was identified on the right side of the gland. It was dissected out. The patient underwent clinical and biochemical remission, but no adenoma was identified on histopathological examination, as it might have been aspirated out due to its small size.Our third patient is a 31-year-old female, a case of primary MRI–Negative Cushing’s disease with central gradient on IPSS. There was suspicious lesion at the junction of the upper end of the pituitary gland and lower end of the stalk on PET-CT and perfusion MRI. Surgical exploration was planned after several rounds of neurosurgery-neuroradiology-neuroendocrine tumor board meetings. She underwent exploration via an extended approach. The sellar floor, turberculum sellae, and the planum sphenoidale were drilled. Dura was opened. We first explored the upper middle part of the pituitary gland but did not find any adenoma. The pituitary stalk was then exposed, but we still did not find any tumor. The diaphragma was cut to expose the junction of the pituitary gland with the stalk. Adenoma was identified at the junction of the gland and the stalk and it was removed. A standard three-layered closure was performed with fat and augmented with glue, as there was intraop CSF leak. The patient underwent remission, and an adenoma was identified on histopathological examination.Our fourth patient is a 29-year-old female, a case of recurrent MRI–Negative Cushing’s disease, operated twice before. She underwent her first surgery at another center, and the second surgery was performed at our center. She underwent remission after the previous two surgeries, and adenoma was confirmed on histopathology with an MIB index of 5%. She underwent reexploration of the gland. Dura was opened. A lateral sellar exploration was performed on both sides and suspicious adenomatous tissue was removed. Sella was explored. No adenoma was identified in the sella, and thus a hemihypophysectomy was performed. Sella was packed with fat and augmented with glue as there was suspicion of an intraop CSF leak. She did not undergo remission, and histopathology did not reveal an adenoma. She was thus managed with a bilateral adrenalectomy.Our fifth patient is a 39-year-old female, a case of primary MRI–Negative Cushing’s disease. On pituitary perfusion MRI there was a suspicious lesion in the floor on the right side. She underwent exploration of the gland via a standard endoscopic endonasal transsphenoidal approach. Dura was opened from 5-o’clock to 7-o’clock position. Adenomatous tissue was seen in the right side of the gland which was removed. Lateral sellar exploration was performed and suspicious tissue was removed. Sella was packed with fat and augmented with glue as there was suspicion of an intraop CSF leak. The patient underwent delayed remission, although histopathological examination did not reveal an adenoma.Our sixth patient is a 45-year-old female, a case of recurrent MRI–Negative Cushing’s disease, operated once before at a different center. She had undergone remission after the first surgery with histopathological confirmation of an adenoma. A suspicious lesion was seen on the stalk on preoperative MRI. She underwent exploration of the gland via an extended approach. The tuberculum was drilled. Calcified tissue from the previous surgery was seen in the sella and was removed. No adenoma was identified in the sella; thus, the stalk was explored. Dura over the planum was cut and normal pituitary stalk was seen. Adenoma was identified on the stalk and removed. Closure was performed using fat and fascia as there was CSF leak intraop. Postop the patient underwent remission, and histopathology revealed the presence of an adenoma. She developed recurrence a year later and underwent bilateral adrenalectomy for the same.Based on our experience, we propose the algorithmic management of MRI–Negative Cushing’s disease. In patients with a negative dynamic contrast-enhanced MRI of the sella with central localization on IPSS, imaging using gallium DOTANOC PET-CT can be used to rule out an alternative source of ACTH secretion. After ruling out the differential diagnosis of ectopic Cushing’s disease, a 3T pituitary perfusion MRI should be performed as an adjunct to localize an adenoma. If an adenoma is identified on these, the patient should undergo exploration via a standard endoscopic endonasal transsphenoidal approach. If an adenoma is not identified on these, exploration of the pituitary gland should be performed via an extended endoscopic endonasal transsphenoidal approach. In case of persistence or recurrence after exploration, repeat imaging should be performed and reexploration should be considered via an extended approach. This surgical exploration is planned after several rounds of tumor board meetings. The gland and the lateral sella should be explored first, followed by exploration of the cavernous sinus or pituitary stalk in case of suspicion of an ectopic tumor in these sites. If still no adenoma is identified intraop, a hemihypophysectomy should be performed. In case of nonremission after reexplorations, the patient should undergo further management with bilateral adrenalectomy or stereotactic radiosurgery or medical management. Thank you.We would like to thank the technical and application specialists from the Neurosurgery Skills Training Facility, Neurosurgery Education and Training School, All India Institute of Medical Sciences, New Delhi, India. We thank Mr. Shashi Shekhar and Mr. Trivender Yadav for their valuable support.Dr. Sadhwani reported a grant (no. BT/PR13455/CoE/34/24/2015) from the Department of Biotechnology, Ministry of Science and Technology, Government of India. Professor Suri reported being a principal investigator during the conduct of the study.Primary surgeon: Suri. Assistant surgeon: Raheja, Bora. Editing and drafting the video and abstract: Suri, Sadhwani, Raheja, Bora, Garg, R Sharma. Critically revising the work: Suri, Sadhwani, Raheja, Bora, Garg. Reviewed submitted version of the work: Sadhwani, Raheja, Bora, Khadgawat, Garg, R Sharma. Approved the final version of the work on behalf of all authors: Suri. Supervision: Sadhwani, Bora, Khadgawat, Garg. Diagnosis: MC Sharma.


A splenic artery aneurysm is considered an abnormal dilatation of the splenic artery layers greater than 1 cm in diameter. First described by Beaussier in 1770, it affects 1% of the population but carries a major risk for life-threatening complications of rupture in 3%–10% of cases regardless of its congenital or acquired etiology. The presentation is highly variable, from asymptomatic incidental discovery during routine imaging to aneurysmal rupture causing acute abdomen, massive gastrointestinal bleeding, and hemorrhagic shock.Herein, we present a 19-year-old male patient who presented with epigastric pain and abdominal rigidity associated with a moderate amount of free peritoneal fluid that was found to be a ruptured SAA after immediate laparoscopy, which was successfully managed with splenectomy.SAAs are a rare etiology of acute abdomen and hemorrhagic shock but have a very high risk of mortality even upon immediate intervention, requiring a very high level of vigilance and a low threshold for surgical intervention in unstable patients presenting with abdominal pain. Although rare, with only 1% prevalence ; howeverA 19-year-old male patient with no significant medical or surgical history presented to the emergency department complaining of a 1-h history of sudden onset severe stabbing epigastric pain that then became generalized, radiated to both shoulders and back, associated with nausea but no vomiting, increased upon movement and while in the supine position but that was not related to food. The patient said they had no history of preceding trauma, vomiting episodes, hematemesis, melena, or changes in bowel habits. He also outlined that he had not had similar previous episodes, postprandial pain, alcohol consumption, and chronic analgesia ingestion. He worked as a construction worker and had no encounters with animals or sick contacts.Upon examination, he was mildly hypotensive with a blood pressure of 106/72 and tachycardic. Capillary refilling time was 2 s; he was conscious and alert, lying in bed motionless in pain. He was pale and sweaty. Abdominal examination revealed voluntary guarding with generalized tenderness maximum on the epigastric area, questionable costovertebral angle tenderness, and hypoactive bowel sounds, and rectal examination revealed no blood or black tarry stools. Chest and heart examination revealed no abnormalities.9/L), a platelet count of 254 platelets per microliter, normal liver enzymes, normal kidney and liver function tests, and normal lipase and amylase levels. Abdomen x-ray showed no bowel dilatation or air-fluid levels. Chest x-ray showed no subdiaphragmatic free air. Abdomen ultrasound showed normal internal organs of the abdomen except for moderate amounts of free fluid and no signs of cholecystitis, biliary dilatation, hydronephrosis, or splenic abnormalities. He underwent contrast-enhanced abdomen and pelvic computed tomography that showed moderate amounts of free fluid around the spleen, in the pelvis, between the stomach and spleen, and in Morison's pouch (en bloc. Running of the bowel and other internal organ examination was also done, and the result was normal with no other abnormalities. Hemostasis was secured, and the patient received two packed red blood cell units intra-operatively. Then, he was transferred to the intensive care unit for close monitoring with no immediate or early complications.Initial laboratory tests showed a hemoglobin level of 14 g/dL, a white blood cell count of 8 WBCs per microliter (8 × 10's pouch , 2; fluiPostoperatively and after resuscitation, hemoglobin decreased to 11 g/dL, but white blood cell count, platelet count, lactic acid, and C-reactive protein were normal. The patient recovered well in 4 days with no events and was discharged home in excellent condition.Haemophilus influenzae vaccines and annual vaccination as scheduled.At 2-week follow-up after discharge, the patient reported no complaints, was vitally stable with normal physical examination, had a hemoglobin level of 12.7 g/dL, and had taken recommended postsplenectomy vaccinations including pneumococcal, meningococcal, and Pathologic examination of the resected specimen showed a 2.5-cm true splenic artery aneurysm with normal histology, excluding secondary pathological causes of the aneurysm.A splenic artery aneurysm is an abnormal dilatation of the splenic artery layers greater than 1 cm in diameter . It was Symptomatic SAAs only occurred in 20% of cases, which presented with epigastric and left upper quadrant abdominal pain. Other nonspecific complaints included anorexia, nausea, or vomiting. The final diagnosis is nearly always obtained by abdominal imaging . UnfortuAnother rare presentation of SAA rupture is the double rupture phenomenon of the SAA that was described for the first time in 1930 also by Bockerman, in which aneurysms initially rupture into the lesser omental sac causing transient tamponade, during which patients are still stable, with mild or no symptoms. Six to ninety hours later, blood overflows into the peritoneum through the foramen of Winslow, leading to sudden hypovolemic shock and vascular collapse .Another very uncommon presentation of SAA rupture causes secondary erosion of the SAA to an adjacent organ, leading to gastrointestinal hemorrhage .Erosion of the aneurysm into the splenic vein also leads to arteriovenous fistula and portal hypertension and rarely results in mesenteric steal syndrome and small bowel ischemia .Ultrasonography, pulsed Doppler, contrast-enhanced computed tomography scanning, magnetic resonance imaging, magnetic resonance angiography, and abdominal aortic arteriography are all radiographic modalities capable of diagnosing an asymptomatic aneurysm. Contrast-enhanced computed tomography scanning is the gold standard in detecting aneurysms and intra-abdominal bleeding. Until now, abdominal aortic angiography, however, is the most valuable modality of choice in localizing the bleeding source and assessing collateral vessels .Regarding management, regardless of their dimensions, all symptomatic SAAs are believed to require treatment; patients with aneurysmal dimensions of 2 cm, pregnant or fertile patients, patients with portal hypertension, or candidates for liver transplantation patients also require management even if asymptomatic .Excision, ligation, or revascularization are surgical alternatives, with or without splenectomy. Either an open or minimally invasive approach could be used. Those who are not candidates for surgery and those in elective settings are typically given the endovascular option, which includes various techniques such as coil embolization, covered stents placement, gluing, plug deployment, and endoluminal thrombin, polyvinyl alcohol, particles, or gel foam injection. However, surgical intervention remains the gold standard treatment in most centers, especially in cases of rupture , 10.This study has described a case of a male patient who presented with acute abdomen, signs of shock, and evident free fluid on imaging that was later found to have a ruptured SAA. The rare etiology and rarer presentation in an unusual gender and age group, highlight the importance of having a very high level of awareness about SAAs as a potential cause of bleeding and a low threshold for more advanced imaging modalities for diagnosing conditions with very high mortality and an even lower threshold for intervention in which mortality rates dramatically increase with every minute of delay.I am grateful for the opportunity to share my medical journey through this case report. It is my hope that by sharing my experience, others may gain valuable insights and knowledge that can contribute to the understanding and treatment of similar conditions. I want to express my sincere appreciation to the healthcare professionals involved in my care, whose expertise and compassion made a significant impact on my well-being. Their dedication and commitment to providing quality healthcare have been truly remarkable. Lastly, I extend my gratitude to the authors and researchers involved in the preparation of this case report for their dedication to advancing medical knowledge. Together, we can contribute to improving patient care and outcomes for future individuals facing similar medical challenges.


Epispadias is a congenital malformation marked by the failure of the urethral bulb to tubularize dorsally. This results in a wide-open urethral plate dorsally. Epispadias is frequently associated with bladder exstrophy-epispadias complex. We report the case of a five-year-old patient who presented in the outpatient department with isolated epispadias without any other associated abnormality.This report aims to document this rare case of isolated male epispadias with incontinence and the success of using the modified Cantwell-Ransley technique for its treatment.  Epispadias is a congenital malformation marked by the failure of the urethral bulb to tubularize dorsally. Contrary to hypospadias, where the meatus lies ventrally on the penile shaft, children with epispadias have a wide and open urethral plate that lies on the dorsum. Isolated male epispadias largely remain rare, with an estimated incidence of less than 1 per 100,000 live births , and ureSurgical management of epispadias can be difficult, requiring significant expertise. Surgical aims include reconstructing the anatomy to provide optimal function and cosmetics for the genitals and urethra . CurrentThe modified Cantwell-Ransley repair technique involves mobilizing the urethral plate, moving it ventrally, and separating the corporal bodies while preserving the attachment of the distal plate to the glans. A reverse-MAGPI  procedure is performed on the distal urethra. The corporal bodies are medially rotated and approximated. The other technique, the complete penile disassembly technique, was described by Mitchell and Bagli in 1996 . This teThis report aims to document this rare case of isolated male epispadias with incontinence and the success of using the modified Cantwell-Ransley technique for its treatment.A five-year-old patient presented in the plastic surgery outpatient department of Holy Family Hospital with complaints of an abnormally located external urethral orifice, micropenis, and urinary incontinence. There was no history of painful micturition, catheterization, family history of epispadias, or any other anomalies. On examination, a single external urethral meatus was found to be located at the junction of the glans and shaft of the penis. Moreover, there was a cleft on the dorsal surface of the glans. The size of the penis was less than normal Figure . The scrUltrasonography (USG) of the abdomen and an X-ray of the pelvis were done. No genitourinary abnormality was detected on the USG abdomen except for the contracted gallbladder. X-ray of the pelvis has revealed pubic symphysis diastasis Figure . A voidiThe patient was prepared for surgery in the usual manner. Epispadias was repaired using the modified Cantwell-Ransley technique. It includes the following steps.1. Degloving of penile skin.2. Separation of both corpora cavernosa, urethra, and the corpora spongiosum Figure .3. The urethral shape was tubularized. 4. The urethra was moved ventrally, and lateral glans wings were created for a natural appearance. 5. Reverse-MAGPI procedure on the distal urethra to refine the tip of the penis and position the urethral opening correctly. 6. Finally, after medial rotation and approximation of the corporal bodies, the skin was closed, with repositioning of the meatus Figure .It was advised not to remove the Foley catheter for two weeks. Postoperatively, the patient complained of supra-pubic pain. The frequency and intensity of pain decreased with time. A follow-up visit was done after two weeks, and the Foley catheter was removed. Meatal position and urination were normal, and the patient did not complain of any incontinence. He had an excellent urinary stream and function Figure .Many techniques are reported in the literature for BEEC reconstruction , with vaHowever, the literature on isolated male epispadias is very scarce, and urinary continence in males as a primary outcome is not reported. This is rather unfortunate as continence is an essential issue in these children. Globally, different centers have mentioned variable continence rates depending on the type of epispadias, ranging from 50% to 90% .Nocturnal continence rates rarely approach more than 50%. The main factors contributing to incontinence include the distorted anatomy of the bladder neck and the posterior urethra, along with histological abnormalities in the roof tissues . ContinePelvic X-ray and cystourethrogram should be done to look for any possible pubic symphysis diathesis and urogenital abnormalities. Postoperatively, pain is a common complication. Patients should be monitored periodically to look for any possible urinary incontinence and possible risk of procedure failure, which may require revision. Bladder spasms following repair are common, and anticholinergic medications should be started. These play a role in managing detrusor relaxation as well as provide a nominal degree of local anesthetic action. In this case of isolated male epispadias, the characteristic outward rotation of the innominate bones resulting in a wide pubic symphysis was appreciated via an X-ray pelvis. If very large, these are repaired with anterior and vertical osteotomies. Various surgical procedures have been reported in the literature to treat this anomaly. However, the repair is far from trivial and can be challenging even in the most experienced of surgical hands. Among the techniques described, the modified Cantwell-Ransley has become the procedure of choice due to excellent cosmetic, functional, and anatomical results in isolated epispadias . Surgery aims to achieve a functionally and cosmetically acceptable penis . It is iConsultation should be obtained with psychiatry and urology/andrology during the transition of care into adulthood for comorbid conditions to be addressed properly.The purpose of this report is to document this rare case of isolated male epispadias with incontinence and the success of using the modified Cantwell-Ransley technique for its treatment. Most cases of epispadias are associated with other serious congenital anatomical anomalies, of which bladder exstrophy occurs most often. Finding isolated epispadias without associated anomalies is rare. However, in an isolated male epispadias case, it is essential to look for associated anomalies.


One of the rare tumors of the salivary gland is known as basal cell adenoma (BCA). Only a small percentage of salivary gland tumors affect the minor salivary gland of the oral cavity while the majority are found in the parotid gland. We present a rare case of BCA involving the left buccal mucosa of a 45-year-old female. Magnetic resonance imaging (MRI) showed well defined solid mass measuring 1.9 x 1.5 cm in the left buccal space inseparable from the buccinator muscle. The T2-weighted image demonstrates a hyperintense signal post-contrast. Ultrasound-guided fine needle aspiration cytology revealed cellular basaloid neoplasm of uncertain malignant potential. Thereafter excision of the mass was performed through a transoral approach under general anesthesia. Histopathology of the mass showed encapsulated basal cell neoplasm in favor of BCA. The patient was doing well after the surgery and has intact facial nerve and adjacent nerves such as the auriculotemporal nerve and great auricular nerve with no complications then she kept on routine clinic follow-ups, and the surgical site recovered successfully. Therefore, we conclude that MRI and biopsy provide useful information to differentiate between benign adenoma and malignant adenocarcinoma. BCA should be considered in a differential diagnosis of an isolated neck mass. Surgical excision demonstrates an excellent prognosis. Salivary glands basal cell adenoma (BCA) is a benign tumor that is rare and has a high risk of recurrence with a generally good prognosis. It’s classified as a separate entity in the classification of tumors of the salivary glands by the World Health Organization (WHO) . SeveralA 45-year-old female not known to have any medical illness was referred to the clinic to evaluate a painless, asymptomatic mass at the left buccal mucosa noticed seven years ago which was small and then had been slowly growing over the past five years. It started off being easily moveable and over time became more fixed. She denied any history of pain, skin discoloration, numbness, facial weakness, xerostomia, difficulty breathing, dysphagia, odynophagia, or other compressive symptoms. A bimanual examination of the oral cavity revealed an intraoral left buccal mass that is round, firm, mobile, and not attached to the skin with no fluctuations. There were no skin ulcers or palpable lymph nodes, and the rest of the head and neck evaluation was unremarkable. Ultrasound-guided fine needle aspiration cytology was performed on the patient, which demonstrated a cellular basaloid tumor with uncertain malignant potential. The head and neck magnetic resonance imaging (MRI) revealed a well-defined solid mass measuring 14x19x15mm  centered at the left buccal space. This lesion is showing an isointense signal on T1, an intermediate signal on T2 with the internal vascular signal void, and homogeneous intense enhancement post-contrast with the impression of low-grade minor salivary gland tumor  in 1972, adenomas of the salivary gland were categorized into two types: monomorphic and pleomorphic adenomas. BCA was classified as a form of monomorphic adenoma in the first version published in 1972 . FurtherHistopathologically, BCA is composed of fairly monotonous and homogeneous cells that resemble basal cells and they are distinguished by the growth of a mass from these cells that resembles a tumor. Depending on the World Health Organization (WHO) classification, basal cell adenoma is categorized into four groups according to the way the adenoma is arranged: solid, tubular, trabecular, and membranous . NeverthDifferentiation between basal cell adenocarcinoma (BCAC) from BCA sometimes is challenging. One of the diagnostic criteria to differentiate between basal cell adenocarcinoma from BCA is the presence or absence of local infiltrative to the adjacent structure. In our case, the tumor was round, firm, mobile, confined, not attached to the skin with intact facial nerve and adjacent nerves such as auriculotemporal nerve and great auricular nerve, lymph nodes and vasculature with no compressive or infiltrative symptoms. There were no skin ulcers or palpable lymph nodes, and the rest of the head and neck evaluation was unremarkable which goes more with BCA rather than basal cell adenocarcinoma . The mosBCA of a minor salivary gland is a rare benign head and neck tumor. MRI and biopsy provide useful information to differentiate between benign adenoma and malignant adenocarcinoma. BCA should be considered in a differential diagnosis of an isolated neck mass. Surgical excision demonstrates an excellent prognosis.