Primary hyperparathyroidism due to parathyroid adenoma commonly causes raised serum calcium and focal giant cell lytic lesions in bones known as Brown’s tumors. It is more common in females in the post-menopausal age group.We report a case of a 29-year-old female patient with Brown’s tumor maxilla in a clinical setting of normocalcemic primary hyperparathyroidism. The patient presented to us with facial and palatal swelling for which FNAC was done. Cytology revealed hemosiderin-laden macrophages suspicious for Brown’s tumor. On further imaging studies such as CT Neck, Tc99 Sestamibi scan, and other biochemical tests like parathyroid hormone assay and serum calcium level, the diagnosis of a hyperfunctioning parathyroid gland with normal calcium level was made. Parathyroidectomy was performed and parathyroid adenoma came out to be the primary pathology. On post-operative follow up there was regression of the swelling on the face and palate relieving the patient symptomatically. The diagnostic suspicion of primary hyperparathyroidism should be kept in mind whenever a young female presents with suspected Brown’s tumor, even with normal serum calcium levels, for appropriate management. Ours was a highly uncommon case that was a diagnostic challenge and had a successful treatment outcome. Very few such cases have been reported in the literature to date to the best of our knowledge. Osteitis fibrosa cystica also known as brown’s tumor is a non-neoplastic, reactive lesion resulting from the excessive osteoclastic activity . Brown’snd case reported. The disease can affect any age group, but it is more common among individuals older than 50 years and is three times more common in women than in men . The terA 29-year-old female came to the ENT department with complaints of swelling over her left cheek for the past 6 months  which waA complete head and neck examination revealed a firm to hard swelling in the left cheek which was approximately 6×5cm in size. On intraoral examination- there was a soft tissue swelling involving the left half of the hard palate and extending up to the soft palate . ContrasCytological aspirate from the swelling revealed features suggestive of Brown’s tumor. Further investigations included intact parathyroid hormone assay, serum calcium and phosphate, serum alkaline phosphatase, serum vitamin D, serum creatinine, and urine creatinine. The intact parathyroid hormone serum level was found to be elevated at 297.7 (15.0-65.0 pg/mL), serum calcium level was normal-9.15 (8.6-10.3 mg/dL), serum phosphate was decreased-2.34 (3.4-4.5 mg/dl), alkaline phosphatase was elevated-360.4 (30-130 IU/L). Other investigations such as vitamin D, serum creatinine, and urinary calcium came out to be normal thus ruling out secondary causes of hyperparathyroidism. On ultrasonography of the neck, a hypoechoic lesion of size 18.3×9.0 mm at the inferior pole of the right thyroid lobe was reported. The patient was then posted for a CECT scan of the neck which revealed an ovoid lesion in the right trachea-oesophageal groove along the posteroinferior aspect of the right thyroid lobe likely a parathyroid lesion . The scaDiagnosis of a parathyroid lesion with hyperparathyroidism with normocalcemic maxillary sinus Brown’s tumor was made. The patient was planned for right parathyroid mass excision. Intra-operatively, an enlarged right inferior parathyroid gland  was encoOne day post-surgery patient developed hypocalcemia which was managed by calcium and Vitamin D supplements. The patient was kept under close observation for early detection of signs and symptoms of hypocalcemia for their timely management. Serial ECG monitoring followed by intravenous calcium gluconate administration was done when needed. The patient was discharged 7 days after surgery with near normal calcium and parathyroid hormone levels (29.39 pg/mL). Post-excision there was a significant reduction in the size of Brown’s tumor thus providing symptomatic relief to the patient . On 11 mBrown’s tumor can be caused by primary, secondary, or tertiary hyperparathyroidism. Primary hyperparathyroidism is usually asymptomatic and its only initial manifestation is hypercalcemia . However rarely a patient can present with normal serum calcium level despite having a deranged parathyroid hormone level and associated skeletal lesions, as in our case. Our patient had no other signs and symptoms of hyperparathyroidism such as nephrocalcinosis, nephrolithiasis, or gastrointestinal and neurological manifestations and Brown’s tumor was the sole manifestation. So, in a patient with osteolytic bone lesions and raised parathyroid hormone assay, serum calcium levels can be normal and further workup should be done to rule out primary hyperparathyroidism. Brown’s tumor is usually seen in females of more than 50 years of age with the mandible being the most common site (However, in our case, a 29-year female presented with maxillary sinus Brown’s tumor. Brown’s tumor can be misdiagnosed as a primary bone tumor on the basis of radiology, so cytology and blood investigations help to rule out other conditions such as aneurysmal bone cyst, osteosarcoma, giant cell reparative granuloma or giant cell tumor (In a case report by Er et al., in a 57 year female with multiple sclerosis, a CT scan along with incisional biopsy, and biochemical investigations were done for diagnosing Brown’s tumor . HoweverIn our patient following parathyroidectomy, serum PTH levels dropped to normal 10 minutes post excision. The patient’s serum calcium level also dropped which was corrected by giving calcium and vitamin D supplementation. The patient is now kept on regular follow-up and is symptomatically improving with an apparent decrease in size of the Brown’s tumors. On 11 months follow-up, the maxillary Brown’s tumor had more than 50% reduction in size with near normal appearance of the patient’s palate. Serum calcium and parathyroid levels were normal after 11 months of follow-up. All osteolytic lesions of the maxilla and mandible do not require primary excision as the treatment of choice. Parathyroidectomy is the mainstay treatment for Brown’s tumor due to primary hyperparathyroidism.Brown’s tumor regress after the management of parathyroid lesions. Even in the setting of normocalcemia, a patient presenting with locally aggressive osteolytic maxillary sinus mass, FNAC, and serum PTH levels should be done to rule out Brown’s tumor.


Rare giant vestibular schwannomas (GVSs) invade the temporal bone extensively, which carries unique risks for surgery owing to their complicated relationship with adjacent structures, difficult dissection of the temporal bone, and high risk of complications. The underlying mechanism of this invasive behavior remains unknown.We report on a 28-year-old woman who presented with typical hearing loss and facial paralysis (House-Brackmann II). Magnetic resonance imaging exhibited a giant mass (∼5.0 cm) in the right cerebellopontine angle (CPA), petrous apex, and middle cranial fossa. Her primary diagnosis was GVS with petrous apex invasion. With the aid of presurgical imaging reconstruction and intraoperative facial nerve monitoring, we adopted a sequential therapeutic strategy, which included microsurgery for the CPA lesion followed by gamma knife radiosurgery (GKRS) for the petrous mass. During follow-up, stable tumor control was achieved with functional preservation of the facial nerve and no other complications. The postoperative immunohistochemical examination demonstrated dramatic intratumoral inflammation, which suggested its potential role in bony erosion. We reviewed the literature of large vestibular schwannoma with a petrous invasion and further discussed its treatment.Microsurgery remains the top therapeutic strategy for GVS. However, gross total resection with functional preservation of cranial nerves is challenging to achieve once the temporal bone is involved. In this case, we applied a planned and sequential approach of microsurgery and GKRS with a promising outcome, which highlighted this combinational strategy in this rare situation. In addition, pathological examination suggested that intratumoral inflammation might play a role in the bony erosion of GVS. Longer observation and more cases are needed to further investigate its molecular mechanism and treatment plan. Vestibular schwannoma (VS) is a primary tumor of the 8th cerebral nerve. VSs account for 8% to 10% of intracranial tumors, and their incidence rate dramatically increased from 3 to 34 cases per million annually over just a few decades . A typicMicrosurgery and gamma knife radiosurgery (GKRS) are the primary treatment methods for VSs. However, the literature regarding therapeutic plans for a GVS with petrous invasion was limited. To the best of our knowledge, no studies have reported a planned and sequential approach of microsurgery and GKRS for this rare condition. In this study, we reported the case of a young woman with GVS and petrous apex erosion. We adopted a sequential approach of microsurgery and GKRS and achieved satisfactory tumor control. Meanwhile, a pilot exploration of intratumoral inflammation was perform in this patient to investigate its role in bony invasion. We further reviewed the literature of large VSs with petrous invasion and discussed its therapeutic strategy.A 28-year-old woman presented with hearing loss for five years, intermittent dizziness and headache for one month, and facial paralysis for eight days (House-Brackmann II). On admission, neurological and physical examination revealed profound mixed hearing impairment of the right ear  and proinflammatory cytokine (tumor necrosis factor-α [TNFα]) was evaluated via immunohistochemistry on 4-μm paraffin slides. For the control, a typical large VS patient was randomly selected from the pathological database. Compared with the control subject (data not shown), obvious higher density of Iba-1 positive cells  was obseThree months after surgery, the patient’s clinical symptoms (dizziness and headache) were relieved, and no residual lesion was observed in the CPA region. GKRS was applied according to plan using the Leksell Model B Gamma Knife  for the tumor in the middle cranial fossa and petrous bone. The maximum marginal dose was 14 Gy with a 50% isodose curve. After the GKRS, there were no new symptoms. One month after GKRS, postoperative MRI demonstrated that the petrous part of the tumor had asymptomatic enlargement and degeneration of its central region . One yeaOur review involved case reports of GVS with the petrous apex invasion. We searched journal articles in PubMed, Embase, and Web of Science from database inception to May 2021 using the terms “Giant vestibular schwannoma”, “Petrous apex invasion,” “Middle cranial fossa” and “Surgery”. The search was limited to articles in English.Four eligible reports (seven cases) were identified  in the lVSs usually originate from the vestibular ganglion and develop at an insidious rate despite their benign nature in pathology. If left untreated, some VSs could grow more than 4 cm in one dimension (GVS) and compress adjacent cranial nerves, vessels, and the brainstem . TypicalMost of the literature regarding GVS was clinical and imaging research. The mechanism of the invasive behavior remains unknown. In a radiological study on the association of IAC widening and tumor characteristics, Takada et al. proposed that the potential mechanism underlying IAC destruction included  repeatedIn the present case, we investigated various factors related to extensive invasion of the temporal bone. However, the patient exhibited no signs of osteoporosis, and her laboratory examinations, including serum levels of calcium, phosphorus, and alkaline phosphatase, were within physiological levels. She also had no history of head trauma, and her preoperative CT scan showed normal pneumatization of the temporal bone. During the surgery, the tumor showed a typical VS pathological appearance. Together with her presurgical imaging, there was no evidence of intratumoral stroke. Her histological findings demonstrated typical benign VS features, and there was no unusual vascularity or any malignant features. Notably, the suspicious soft tissue in the mastoid cells detected in CT scans was confirmed to be part of the tumor during the surgery, which suggested that the lesion eroded the cortex of the IAC and invaded the bone through the pneumatised tract.Per established study in diseases related to common bony destruction, such as rheumatoid arthritis and bone metastases, systematic and regional inflammation could lead directly to this bony change via promotion, differentiation, and maturation of osteoclast . OsteoclFor neurosurgeons, GVS carries unique risks for microsurgical resection, owing to their large dimensions, deep location, and complicated relationship with adjacent vessels and nerves. GVS invading the petrous apex is extremely rare, making it impractical for resection alone, owing to its high risk of severe complications and treatment failure . The lim3), transient tumor expansion after surgery, large dose radiation, and secondary intratumoral and peritumoral edema. could lead to high occurrence of late complications, e.g., osteoradionecrosis, fibrosis, trismus, and etc.  Thus, loGVS with extensive temporal invasion is rare, and microsurgery remains the primary therapeutic strategy. However, gross total resection with functional preservation of cranial nerves is not easy to achieve. In this case, we applied a planned and sequential approach of microsurgery and GKRS with a satisfactory outcome, which highlighted this combinational strategy in this rare situation. In addition, pathological examination suggested that intratumoral inflammation might play a role in the bony erosion of GVS. Longer observation periods and more cases are needed to further investigate its molecular mechanism and treatment plan.


Acute epiploic appendagitis is a rare cause of abdominal pain, often misdiagnosed as acute appendicitis or diverticulitis given similar clinical presentation and findings. The treatment is supportive care and is typically self-limited. The osteopathic structural exam can give insight into pathology and in this case, was suggestive of a non-appendiceal origin of her pain, in which emergent surgery could be avoided. Requiring computerized tomography to identify, acute epiploic appendagitis is a rare cause of abdominal pain and should be considered in the differential diagnosis. Abdominal pain is a common complaint seen in outpatient primary care and inpatient emergency medicine alike. The differential diagnosis is broad, and it can have benign to life-threatening etiologies, ranging from gastroenteritis to acute surgical abdomen . DependiOsteopathic physicians are trained to integrate the medical history of a patient with palpatory examination through the osteopathic structural exam which allows the expansion of differential diagnoses and the consideration of viscerosomatic dysfunction for the localization of symptoms . Here weMs. L is a 54-year-old Latinx female who had presented to the urgent care for four days of diffuse abdominal pain, which had localized to the right lower quadrant earlier that morning. Past medical history was significant for hypertension, hyperlipidemia, and anxiety. Surgical, family, and social history were noncontributory. She denied a prior history of similar abdominal pain, sick contacts, new sexual partners, hematemesis, hematochezia, or changes in urinary/bowel movement. She notes that pain was exacerbated with activity, deep inspiration, and yawning, without any alleviating factors.Vitals were unremarkable as the patient was afebrile, normotensive, normocardia, and saturating 99% on room air. Physical exam showed decreased bowel sounds on abdominal auscultation, with rebound tenderness and guarding on palpation. Provocative measures such as the McBurney’s point, psoas sign, and obturator test were equivocal. The point-of-care pregnancy test was negative. She was sent to the emergency room due to concern for acute appendicitis, in which abdominal ultrasound was negative. The point-of-care ultrasound was not available and the patient was sent to the emergency room for further evaluation.Upon admission to the emergency department, the patient was given a 30-milligram ketorolac injection for pain as it had significantly worsened in the right lower quadrant. Blood work was unremarkable, with electrolytes within normal limits and mild leukocytosis with a white blood cell count of 11.4 . CT of the abdomen revealed antimesenteric stranding adjacent to the ascending colon, with central fat attenuation, most compatible with epiploic appendagitis (Figure Outpatient follow-up two days later revealed resolving symptoms and an osteopathic structural exam was performed Table . On examEpiploic appendages are small outpouchings of serosa present on the external surface of the colon that project into the peritoneal cavity . ServingThis case represented a typical case of epiploic appendagitis, however, it also serves as a reminder of the value of the osteopathic structural exam in localizing and aiding in the diagnosis of pathology. Viscerosomatic reflexes, in which visceral and somatic pain afferent nerve signals overlap in the dorsal horn and influence each other, can present as skin erythema for acute processes versus skin blanching for chronic states . DependiManagement is supportive of nonsteroidal anti-inflammatory drugs and is self-limiting as symptoms resolve in 3-14 days . The risEpiploic appendagitis is an uncommon cause of right lower quadrant pain and should be considered when developing a differential diagnosis. The clinical course is typically self-limited and resolves with pain medication. The osteopathic structural exam can give insight into ruling out other pathologies such as acute appendicitis, however, imaging modalities such as ultrasound and CT are needed for definitive diagnosis. Greater research is needed in correlating osteopathic findings to physical exam findings and pathology.


Parosteal lipoma is a rare and benign neoplasm originating from mature adipose tissue near the periosteum. Clinically, it is difficult to diagnose due to its similarity to the clinical manifestation of sarcoma, so imaging, histopathology, and immunohistochemistry examinations are necessary.A 54-year-old woman presented with lump on the right thigh that had gone through surgery eight years prior, with a diameter of 20 cm, with a partly hard and partly soft consistency, the patient was diagnosed with suspected recurrent liposarcoma. We performed wide excision and histopathological results showed a proliferation of bone cells and cartilage cells that were lobulated, surrounded by a proliferation of fat cells with no pleomorphism or immature cells.Parosteal lipomas are neoplasms derived from adult adipose tissue, usually connected to the periosteum, and rare and benign. Two theories of pathogenesis of parosteal lipomas. (1) tumors arise from the differentiation of stem cells derived from adipose tissue, (2) the tumor is derived from secondary metaplasia of fibroblasts due to recurrent trauma, metabolic changes, or ischemia. Based on the theory, it is likely that in this case is due to the presence of differentiation of adiposa tissue due to the non-acquisition of a history of trauma.Parosteal lipoma is a rare case of benign neoplasm, which is difficult to diagnose clinically due to its similar sarcoma, thus requiring imaging and histopathological examination. The treatment of choice is wide excision by taking the entire tumor to prevent a recurrence. •45 year old woman with recurrent parosteal lipoma.•The case of parosteal lipoma is a benign tumor.•A neoplasm originating from mature adipose tissue, mostly located in the bone cortex below the periosteum.•The incidence is 0.3% of all types of lipomas and 0.1% of primary bone neoplasms. Microscopically, parosteal lipoma appears as bone and cartilage against a background of predominantly mature adipose tissue . Clinica2A 54-year-old housewife with a lump on the right thigh. The patient was referred from a peripheral hospital, with a history of eight years ago there was a lump in the same place as the diameter 5 cm, a surgical removal was performed by a general surgeon with the results of the pathological anatomy of the lipoma. The patient reported having two lumps with a diameter of 20 cm; The first lump is located on the lateral side with a soft consistency, while the second lump is located on the medial side with a hard consistency. The patient does not feel pain or burning sensation in both lumps. There is no history of trauma, and there is no family history with cancer.The results of MSCT with contrast showed solid and some semi-solid lesions, which consisted of calcifications and fat at the one-third proximal to one-third distal of the femur. The lesions had smooth borders with partly irregular and septated edges with the size of 23 × 16 × 27 cm.At Dr Moewardi Hospital, our surgery is performed in a team, an oncology surgeon as an operator with anesthesia performed regionally. The patient's position is tilted to the left, the lump is located above. We perform a wide excision, obtaining an intermuscular encapsulation mass with a soft consistency in the lateral position and a hard surface on the medial part resembling a bone. All lumps were successfully removed. Post surgery is not found interference in carrying out activities.The microscopic examination showed the proliferation of bone cells and cartilage cells, which were lobulated, surrounded by proliferating fat cells. There were no signs of malignancy. As for the immunohistochemical examination, S-100, vimentin, SMA, and HMB-45 showed positive results.3Dr. Seerig first discovered parosteal lipoma in 1836. It is a neoplasm originating from mature adipose tissue, usually connected to the periosteum, mostly located in the bone cortex below the periosteum, and is rare and benign. The incidence is 0.3% of all types of lipomas and 0.1% of primary bone neoplasms. The term parosteal lipoma was first made famous by D'Arcy Power in 1888, which implies the relationship between the tumor and the bone while not always originating from the bone (periosteum) . ParosteParosteal lipoma consisting of fat tissues and some differentiation of bones and cartilage is usually located inside the subcutis fat or in the muscles ,7. ThereDebras et al. reported a patient with a parosteal lipoma on the leg. Physical examination showed a painless, mobile, and slowly growing mass with a hard consistency, with a firm border, of similar color to the skin or the surrounding mucosa, without any signs of inflammation, no signs of limitations in the range of motion (ROM) ,8,9,10.In our case, the lump was found on the femur. The lump made an impression of two separate masses, in which the lateral lobe had a more superficial tumor with a soft consistency. In contrast, the lump at the medial was located deeper and closer to the femur and was hard inconsistency. With no ulceration, tenderness, swelling, erythema, or another skin lesion, the mass slowly grew. The most prominent finding of this case was the history of surgery at the exact location 8 years prior. The history of surgery at the same site and the hard consistency of the lump were the reasons why the lump was initially clinically diagnosed as recurrent mixed liposarcoma.Parosteal lipomas are difficult to distinguish from parosteal sarcoma, osteochondroma, liposarcoma, lipoma, and chondrosarcoma, clinically may appear with similar clinical manifestations. In our case, clinically the lump is more similar as a mixed liposarcoma. based on its prevalence, recurrency of parosteal lipoma does not occur after intoto-excision of the tumor, while for liposarcoma, there is still a 33.8% chance of recurrence .Based on the classification of soft tissue tumors by WHO 2002, conventional lipoma consists of mature adipose tissue. It is located in the subcutaneous region  or inside the soft tissue (lipoma), or even on the surface of the bone . Intraosseous lipoma is located inside the bone. The terms osteolipoma and ossifying lipoma are currently used for soft tissue tumors with no relation to the periosteum. In contrast, parosteal lipoma is used for tumors with relation to the periosteum .The microscopic examination found partly encapsulated lesions mainly consisting of lobules of univaculated mature adipocytes and bone trabeculae, with evenly spread lands of benign cartilage. Between the adipose tissue, proliferation of fibroblast, chondrocyte, osteocyte, and blood vessels with thick walls could be seen. Osteocytes and osteoclasts surrounded the bone trabeculae. Mixed liposarcoma is a malignant tumor consisting of circle to oval-shaped primitive mesenchymal cells and a variable number of small signet ring lipoblasts in the myxoid stroma. Liposarcoma differentiates well and is also known as an atypical lipomatous tumor. It is a locally aggressive and malignant mesenchymal neoplasm composed of mature adipocytic proliferation, which exhibits significant malignant cytology; it may occasionally show areas of chondroid metaplasia without extensive myxochondroid matrix . In thisImmunohistochemical staining of liposarcomas showed that the more mature the adipoblast differentiation, the higher the positive level of vimentin and S-100 expression. Moreover, the nuclear proliferation index was <1%, and the Ki-67 proliferation index was low. KP1, LAM, CD68, and CK showed positive results in some cases, while EMA, SMA, MSA, HMB45, and GFAP would give negative results . In thisThere are two main theories for the pathogenesis of parosteal lipoma. (1) These tumors may arise from multidirectional differentiation of stem cells derived from adipose tissue, or (2) the tumors originate from secondary metaplasia of fibroblasts due to repeated trauma, metabolic changes, or ischemia. Several cytogenetic studies on osteolipomas and parosteal lipomas have shown similarities to the t3; 12) translocation in conventional soft tissue lipoma. By identifying the HMGA2-LPP fusion transcription in both the fibro-adipose component and the chondro-osseous component of tendon lipomas, it revealed that the HMGA2-LPP fusion protein induces fibroadipogenesis and osteochondrogenesis [ translocLipomas found in adults often consist of chromosome 12q13∼15 aberration, which causes rearrangement in the HMGA2 gene at the 12q14.3, with breakpoints occurring inside or outside the gene. Panagopoulos studied eleven lipomas and one osteochondrolipoma with a new recurrent chromosomal aberration, t . The molecular study conducted on eight tumors showed three cases expressing full-length HMGA2 transcripts and five cases with chimeric HMGA2 transcripts. Of the three lipomas and osteochondrolipoma, exons 1–3 of the HMGA2 was coupled to the SETBP1 sequence at 18q12.3 or the intragenic sequence of 18q12.3, about 10 kbp distal to SETBP1. In other lipomas, exons 1–4 of HMGA2 fuse to the intronic sequence GRIP1, which maps to the band chromosome 12q14.3, distal to HMGA2. Subsequent HMGA2 fusion would code for the transcript of a putative protein suspected of containing an HMGA2 amino acid residue corresponding to exons 1–3 (or exons 1–4 in one case) followed by an amino acid residue corresponding to the fused sequence.a)X-ray and Computed Tomography scan (CT scan)Further workup for parosteal lipoma includes:Type I. Lucent lesion with dense fat directly adjacent to the radial cortex with no relations to bone changes. Type II. A lenticular mass lesion with a smooth surface of fat density bordering the long midshaft segment. Type III. The AP radiograph depicts a low protrusion of elevation of the medial femoral diaphysis with irregular bone density mixed with areas of fat density. Type IV. Direct imaging shows irregular protrusion of bone density in a density of oval fat.According to Miller 1992, plain X-ray examination of parosteal lipoma in long bones could be classified into 4 types. b)Magnetic Resonance Imaging (MRI)CT scan plays a crucial role in evaluating parosteal lipoma and is useful for documenting the presence of fat and bony elements. In demonstrating the presence of bony elements and evaluating the relationship of the mass to the surrounding bone structures (erosion and/or continuity), a CT scan is the procedure of choice. 3D reconstruction does not add much-needed information, but it may help localize the tumor and demonstrate the anatomical orientation of the mass, a parameter that facilitates excision. In addition, a CT scan provides a fine picture of scattered calcifications. On X-ray and CT scans, parosteal lipoma appears as a calcified soft tissue mass ,8,9,10.bAlthough CT scan is useful in visualizing fat and bony elements, MRI is considered the imaging modality for lipomatous tumors. MRI allows the identification of lipomas, which has a homogenously identical signal intensity to that of subcutaneous fat in all pulse sequences, whereas ossification, calcification, and fibrous connective tissue appear as low signal intensity ,9.The imaging findings show chondroid lipoma, fatty tumors with chondro-osseous differentiation such as lipomas or liposarcoma, tumors including a myxoid component such as myxoid liposarcoma and extraskeletal myxoid chondrosarcoma, should also be included in the differential diagnosis. Both lesions may show heterogeneous signal changes on MRI associated with varying amounts of fat and chondroid tissue. A small number of a reticulated or linear pattern of mature fat and a cyst-like appearance found in myxoid liposarcomas can help differentiate them from chondroid lipoma. Its invasive nature can also help differentiate extraskeletal myxoid chondrosarcoma from chondroid lipoma . Due to The treatment of choice for parosteal lipomas is excision of the entire tumor ,7. DurinThe prognosis for parosteal lipomas is the same as for other lipomas, and the tumor usually does not recur after complete excision . Evaluat4Parosteal lipoma is a rare and benign case of neoplasm. It is difficult to diagnose clinically due to its similarities to the clinical manifestations of mixed liposarcoma, so imaging and histopathological examinations are needed. The treatment of choice for this tumor is wide excision, removing the entire tumor in toto to prevent a recurrence.The patient gave consent to the author for publication.The authors received no financial support for this article's research, authorship, and publication.Informed Consent in writing is obtained from the patient for the publication of the report of this case and the accompanying images.Widyanti Soewoto M.D., Ph.D.: study concept, data analysis and writing the paper. Brian Waskita M.D., Ph.D.: interpretation of anatomical pathology. Amriansyah Pranowo Imamsoedjana: data analysis.1. Name of the registry: Research registry.2. Unique Identifying number or registration ID: researchregistry8022.3. Hyperlink to your specific registration (must be publicly accessible and will be checked):Widyanti Soewoto M.D., Ph.D.Not Commissioned, Externally Peer-Reviewed.The authors declare no potential conflicts of interest.


Elastofibroma dorsi (ED) is a rare tumor that most often occurs in the subscapular and infrascapular region between the thoracic wall, serratus anterior, and latissimus dorsi muscle. Based on a review of the literature, ED has been deemed an extremely rare entity. However, the incidence may be greater and is difficult to determine as the majority of ED being asymptomatic and therefore undiagnosed. Surgical excision is commonly performed when patients present with pain associated with ED. This being the case, it is important to evaluate the factors contributing to the pain seen in these patients and to evaluate the risks vs benefits of intervening in symptomatic ED patients who present for possible surgical intervention. We herein report a case of bilateral ED, situated in the upper back with only the right side being symptomatic in a 56-year-old male laborer. Due to pain in the right upper back, the patient underwent surgical removal of the ED. The postoperative course was uneventful and the patient had an excellent recovery. A review of the literature showed no correlation between pain on presentation and tumor size or location. Major complications of treating these patients include seroma or hematoma formation which according to the literature can be avoided using postoperative tube drainage and compressing bandages. Elastofibroma dorsi (ED) is a rare tumor that most often occurs in the subscapular and infrascapular region between the thoracic wall, serratus anterior, and latissimus dorsi muscle . ED has We herein report a case of bilateral ED, with only the right side being symptomatic, in a 56-year-old male laborer who underwent surgical excision on the right side of the upper back. We performed a literature review to investigate the potential factors that cause ED to be symptomatic and discuss potential surgical interventions given the risk for complications.A 56 y/o male presented with a painful right upper back mass which had gradually enlarged over the past two years. He had been experiencing a sharp non-radiating pain at the site of the mass, which he rated as seven out of ten on the pain scale. The patient had a previous history of a back injury that caused radiating back pain. He had no history of recent/new injury at the time of his presentation. The pain at the site of his mass was different from his chronic pain. The patient reported frequent manual labor at his job as a construction worker that continuously involved heavy lifting.On physical exam, there was an 8 cm mass on the right upper back which was tender to palpation. The patient had no neurological signs. Range of motion (ROM) of upper extremities and shoulders remained intact bilaterally, with some mild discomfort on abduction and adduction of the right shoulder.CT scan of the chest with IV contrast showed bilateral soft tissue masses present in the upper back immediately deep to the trapezius and latissimus dorsi muscles and posterior to the serratus musculature Figure . On the Given the patient’s persistent right-sided pain, lack of relief from over-the-counter medications, and the nature of his job, the patient requested surgical excision of the right-sided mass to relieve his pain and discomfort. Surgical excision was performed under general anesthesia. The mass was found located deep to the deep fascia and was noted to be intramuscular, measuring approximately 6 x 6 cm. Prior to closing the wound, meticulous hemostasis was obtained, and a Blake drain was placed deep in the intramuscular cavity to prevent seroma or hematoma formation The wound was closed in three layers. The specimen was sent for pathological evaluation. The pathology report stated that the lesion was found to be composed of fibrous tissue which showed a bland spindle cell component admixed as well as some adipose tissue. Trichrome stain showed extensive collagen deposition and elastic tissue stain demonstrated dense irregular clumps of elastic material. The specimen was strongly positive for vimentin, with smooth muscle actin, desmin, and s100 being negative. CD34 staining showed some positive cells and p53 staining showed some weak positivity. Based on these findings, pathology determined that these features were consistent with the clinical presentation of Elastofibroma, and no malignancy was identified.Following discharge, the patient was seen on postoperative day six. The output of the patient’s Blake drain was consistently under 25mL per day since surgery and appeared serosanguinous. There were no signs or symptoms of post-operative infection. The Blake drain was removed during that visit. At the patient’s six-week follow-up, his surgical incision had healed well, and he had returned to his regular activities and work without restriction.The PubMed Database was utilized to review previously published case reports and case series of ED focusing on tumor size vs symptoms and the number of cases with postoperative complications. Six individual case reports were included in our analysis, all of which reported pain on presentation with tumor size ranging from 5 x 5 cm to 13.5 x 8.7 cm , initially described as rare, was first reported and named by Jarvi et al in 1961, and was later classified as a benign fibroblastic/myofibroblastic tumor in 2000 by the World Health Organization . ApproxiSince forceful and repetitive movement of the shoulder girdle seems to be involved in ED pathogenesis, an investigation has been done looking at the relationship between manual\heavy activities and the development of ED which has shown a variable correlation of 15% to 95%, with male patients appearing to be more affected due to higher involvement in manual\heavy activities compared to females (71% and 30% respectively) . In two In cases of symptomatic ED, pain is the most common presenting chief complaint, with presentation ranging from mild to severely disabling pain. It has been assumed that the severity of the pain is dependent on the size and location of the tumor. However, based on our review, there was no clear correlation between tumor size and symptoms, with very small tumors presenting with pain and very large tumors remaining asymptomatic in some instances. Also, there was very little variation in the tumor location from our review with the majority of the tumors located in the subscapular region between the serratus and latissimus muscle. There is very little information from the existing literature on the factors contributing to painful ED. Further studies should be performed to evaluate the factors causing ED to become painful. -9.The treatment of ED remains controversial. Given the benign nature of the lesion, surgery is reserved for symptomatic ED. The decision to excise an ED should be made jointly by the patient and surgeon. Those with significant symptoms should be offered an excision and be made aware of the risks of surgery . The mosFurther investigations are needed to determine the factors contributing to pain in patients with ED since the size of ED does not appear to correlate with symptoms. Given that the rates of postoperative complications and recurrence are low, we recommend surgical excision be considered in all patients presenting with painful or symptomatic ED.


Azygos vein aneurysm (AVA) is a rare thoracic pathological entity that mimics a posterior mediastinal mass. However, the pathogenesis of primary azygos vein aneurysms is not clear and its pathology is still being discussed. Some of the AVA are asymptomatic and usually discovered accidentally by routine physical examination.We report the case of a 37-year-old woman who had an azygos vein arch aneurysm with no obvious clinical symptoms. With the analysis of clinical features of the case and AVA morphological characteristics, the AVA was found by a chest computed tomography. Then, enhanced chest computed tomography showed a soft-tissue mass (4.9 × 3.7 × 3.2 cm) in the right posterior mediastinum, which was connected to the superior vena cava and significantly enhanced with contrast agent stratification. The density of the tumor in the delayed stage was the same as that in the azygos vein. The patient underwent video-assisted thoracoscopic surgery. Histopathological evaluation of the surgical biopsy specimen proved to be a completely thrombosed aneurism of the azygos vein arch.AVA is a rare pathology that must be taken into consideration during the differential diagnosis of right posterior mediastinal masses. Thoracoscopic surgery is one of the most preferred treatment options for azygos vein aneurysm.The online version contains supplementary material available at 10.1186/s13019-022-01908-5. Azygos vein aneurysm is an extremely rare cause of the mediastinal mass that typically detected incidentally in asymptomatic patients by the chest radiograph or computed tomography . In geneA 37-year-old female was admitted for surgery for sebaceous cyst of the back with no specific underlying disease or symptoms such as dyspnea, cough, chest pain and so on. The pre-operative chest computed tomography revealed a 4.9*3.7*3.2-cm sized well-defined homogenous opacity in the right lower trachea while her physical examination and hematologic test results were normal. Dynamic contrast enhanced multidetector CT of the patient’s chest confirmed a slowly enhancing mass in the pathway of the azygos venous arch. In addition, there seemed to be a blood shadow during coronary reconstruction. The patient was submitted to surgery and thoracoscopic approach to the resection of the tumoral mass by the operation of video-assisted thoracic surgery during which there displayed a mass arising from the azygos venous arch, which was an enlarged abnormally dark purple cystic lesion with slightly adherent to the surrounding tissues Fig. . FinallyAVA is a greatly rare cause of the posterior mediastinal mass with some symptoms presented depending on the aneurysm size and complications. There have been reports of cases of AVA presenting with some symptoms such as back pain, chest tightness, dyspnea, coughing and orthostatic hypotension . HoweverThe etiologies of AVA have not been clarified, which are divided into idiopathic, acquired, or traumatic  at preseAzygos vein aneurysms must be taken into consideration during the differential diagnosis of right posterior superior mediastinal masses. In view of the diverse clinical presentation, the diagnosis of azygos vein aneurysms is often confused with lymphomas, bronchial cysts, fibromas, and paragangliomas –7, 22. TAVA is a relatively benign condition and its therapeutic strategy is still contentious. At the same time, there are no clear guidelines or therapeutic program for conservative, surgical or endovascular treatment. The aim of any therapy in patients with an AVA is to prevent rupture of the aneurysm, thromboembolism or pulmonary artery hypertension, as well as symptoms caused by compression of adjacent structures there , 14. YetIn conclusion, AVA is a rare lump-like lesion of the mediastinum, which needs to be included in the differential diagnosis. The imaging findings combined with enhanced multidetector CT and three-dimensional reconstruction are useful in establishing the nature of the mass and the origin of vascular lesions to characterize azygous vein aneurysm. Once AVA with thrombosis or symptoms, surgery is recommended and must be performed. If the preoperative diagnosis might be hard to be established, we should consider avoiding the risk of bleeding caused by puncture biopsy and performing an exploratory thoracoscope.Additional file 1. Contrast-enhanced CT of the patient's chest before surgery, 3D reconstruction of the aortic and azygos vessels.Additional file 2. Non-enhanced CT of the patient's chest before surgery.


Solitary Peutz-Jeghers-type polyp (SPJP) is a rare hamartomatous lesion. It is considered as a different entity from Peutz-Jeghers syndrome despite similar histopathological findings. It can be found in the GI tract but rarely in the jejunum. Jejunal SPJP are susceptible to necrosis, ulceration, and intussusception, resulting in GI bleeding or small bowel obstruction.We describe a case of subacute gastrointestinal bleeding secondary to jejunal SPJP to share our approach to this challenging case using therapeutic endoscopyAn 81-year-old male patient with a history of atrial fibrillation on warfarin with stable therapeutic INR levels, who presented with 1-week history of melena, generalized fatigue and shortness of breath on exertion and was found to have profound iron deficiency anemia. Esophageal gastroduodenoscopy and colonoscopy failed to identify the source of bleeding; however, double balloon enteroscopy detected a 4 cm polyp with stalk in the proximal jejunum. Endoscopic polypectomy was performed and the whole polyp was removed. Histopathological examination was consistent with Peutz-Jeghers polyp. The oropharyngeal and skin examination was negative for mucocutaneous hyperpigmented macules and the genetic analysis was negative for STK11 mutation. Follow up magnetic resonance enterography and video capsule endoscopy did not reveal any other polypoid lesion in the GI tract. The patient’s symptoms resolved gradually, and his hemoglobin level returned back to normal levels within 6 months.To our knowledge this is the first case of endoscopic polypectomy during balloon-assisted enteroscopy for jejunal SPJP.ConclusionSolitary Peutz-Jeghers hamartomas are a rare occurrence but should be part of the differential diagnosis for abdominal pain from intussusception, or small bowel bleeding even in elderly patient. Balloon assisted enteroscopy is a valuable technique for the management of solitary small bowel lesions.NoneNone DeclaredLIVER AND BILIARY DISEASE


In its complex way of embryonic evolvement, the diaphragmatic membrane can be involved with various disorders that may partially or entirely not develop. Agenesis of the diaphragm is the term that refers to this maldevelopment. It is the more severe form of congenital diaphragmatic hernia in which intra-abdominal viscera protrude into the thoracic cavity, causing respiratory and gastrointestinal problems. Most neonates delivered with diaphragmatic agenesis do not live more than hours to days of the severity of lung immaturity. However, less than 20 affected cases have been reported so far that survived to childhood and even their adulthood period treated surgically or conservatively. We have reported a case of neglected left hemi-diaphragmatic agenesis for more than six decades, then reviewed all adult diaphragmatic agenesis cases available in the literature for 74 years.A 65-year-old female complaining of worsened dyspnea during the last four months, a chronic history of short breath since her fourth decade of life, and recent surgery with the diagnosis of a diaphragmatic hernia, underwent the thoracotomy twice in which unilateral diaphragmatic hernia was diagnosed then repaired.For the recurrence of her symptoms, she underwent a second thoracotomy, in which the final diagnosis of left hemi-diaphragmatic agenesis was made. A dual mesh patch constructed the defect. The post-operation period was uneventful. We only found 17 cases of adult hemi or bilateral agenesis of the diaphragm reviewing the main medical literature such as Medline and Web of Science. The conservative and operative treatment managements were equal for eight patients in each of them. One of them refused therapy, and one was non-declared in the study. As in congenital diaphragmatic hernia, the most typical side was the left in 10 out of 18. The most complaints patients had followed by coughing and bowel obstruction was dyspnea and dyspepsia.Near the total of the diaphragmatic agenesis cases die in the neonatal population; remained undiagnosed or during an autopsy found. However, typically rare in the adult population, respiratory and digestive disorders are the most prevalent. It is difficult initially because diagnosing is intraoperatively, and no modality is available to help the examiner physician diagnose perinatally.  •Diaphragmatic agenesis is a rare anomaly seen usually in the neonatal period.•The diagnosis is made during autopsy or surgery.•The most common affected side is the left, and bilateral ageneses are reported only once in the literature.•Who survive are usually not symptomatic until adulthood when bowel obstruction, pneumonia, or shortness of breath occur.•We have only found 18 adult diaphragmatic agenesis cases for 74 years now. As cong2The patient was an overweight 65-year-old female from a low socioeconomic status who complained of exacerbated dyspnea during the last four months and was presented to the thoracic surgery centre. Having no pieces of evidence of chronic obstructive pulmonary, any proven cardiopulmonary diseases, or bowel obstruction history but only occasional constipation; however, she has been suffering from activity-related dyspnea since her third decade of life. She took only herbal laxatives and did not have trauma or prior chest surgery. The familial history from the point of any cardiothoracic disorders was unremarkable too. The grade of the chronic difficulty in breathing according to the modified medical research council (mMRC) dyspnea scale was +1. However, she was coerced into referring to a pulmonologist as the scale advanced to +3 in this period. During a physical examination, although not cyanotic, she complained incessantly of being suffocated. The oxygen saturation level was in a normal range. Notably, besides tachypneic clear respiratory sounds at the right hemithorax, bowel sounds were heard from the base to the half of the left hemithorax with decreased left-sided air entry. Blood gas analysis showed only mild respiratory acidosis with some degrees of metabolic compensation. The routine haematological and biochemical analysis gave normal results. Upright chest radiography revealed gaseous shadows suggestive of gastrointestinal components' entrance into the left hemithorax compatible with the left diaphragmatic hernia at first sight .Fig. 1PlThus, the patient with the preoperative diagnosis of left unilateral diaphragmatic herniation underwent surgery by a general surgeon. The complaint disappeared for about four months while the initial symptoms reoccurred. Renewed evaluations such as chest X-rays were representative of recurrent hernias at first sight . An axiaExtubating in less than 24 hours, her respiratory discomfort improved. The postoperative recovery and follow-up durations have been uneventful for two years now.3In its complex way of embryonic development, the diaphragm, the main fibromuscular contractible organ of ventilation, can be involved with various disorders in which the respiratory and digestive systems are also affected. The four major embryologic fountains—the septum transversum, pleuroperitoneal membrane, dorsal oesophagal mesentery, and the body wall-join in developing this dome-shaped septum that separates the abdominal and thoracic cavities around the third-fourth month of gestation . During CDH is the most well-known developmental diaphragmatic disorder due to a failure of the embryonal posterolateral pleuro-peritoneal folds to migrate anteriorly, separating the thoracic and abdominal cavities. Its prevalence varies and is estimated at less than three per 10000 births, according to the reports . CDH hasDA was initially described by Gregers Thomsen as a primordial defect in the development of the lung, corresponding to similar defects in other organs in a thirty-six-year-old woman complaining of shortness of breath and cyanosis after physical activities . We haveAlmost always, agenesis leads to the death of the patients in their first hour or days of life, while those who survive to adulthood are not symptomatic until their third decade ,28. The Pre-operating Diagnosis of DA is almost impossible. Thus, whether the diagnosis is intraoperatively is done mainly for bowel obstruction in diaphragmatic hernia or during autopsy. Although sporadic, genetic consultation is recommended for future pregnancies in the cases of CDH or DAs leading to neonatal death. Bilateral DA is a much rarer entity than bilateral CDH.Answering the question of when to repair the agenesis of the diaphragm is the basis of surgically. Why so? For bowel incarceration and strangulation risk in cases of partial defects or smaller size, immediate repair is unanimously suggested, even in asymptomatic patients . Of courCardiopulmonary function and associated anomalies are mainly the predictor factors for the affected patients' survival rate rather than the defect's size.Delayed surgery and postmortem diagnosis made biases in both series that are divergent criteria for identifying and interpreting the results. Still, certain is that DA is more fatal than CDH.4In the opinion of most of the literature, DA is settled at the end of the spectrum of CDH as the most severe form of it. On the other hand, the larger the defect, the poorer the prognosis. Most neonatal cases die in the first hours and days of life because of corresponding lung hypoplasia or pulmonary hypertension. Those who reach adulthood are not severely symptomatic until adulthood when shortness of breath or even bowel obstruction persuades them to a medical centre. One-third of cases reported were in their third decade of life. All of them presented with respiratory disorders. Therefore, it could be concluded that breath-related problems outburst earlier.Furthermore, those who had tolerated lung-related problems until their middle ages had been referred with digestive complaints of dyspepsia or even intestinal obstruction. It is difficult to diagnose DA intraoperatively, and no modality is available to help the examiner physician diagnose perinatally. Although so rare, numerous adult cases probably did not undergo surgical treatment or were misdiagnosed as diaphragmatic hernias, so the number of reports in the literature is such few that it does not reach 20.The ethics committee of Ardabil university of medical sciences has proved and observed all aspects of this research from proposal to the final form of the manuscript. If necessary, the corresponding author can submit the certificate.We authenticate not receive any financial support.Iraj Feizi was the surgeon who operated on the patient, suggested publishing a case report. Ali Samady Khanghah, a member of the research committee of the hospital prepared the manuscript, decided to advancing it to a “case report and literature review” manuscript, and then pursuits the submission process.1.Name of the registry:Agenesis of the Left Hemi-diaphragm, the Underlying Cause of a Neglected Dyspnea in a 65-Year-Old Female; Case Report and Literature Review2.Unique identifying number or registration ID:researchregistry83193.https://www.researchregistry.com/browse-the-registry#home/registrationdetails/6324d4e828d6cb00212005f4/Hyperlink to your specific registration (must be publicly accessible and will be checked):Ali Samady Khanghah accepts full responsibility for the work and approves the whole process from designing the study to publish.The consent in which the patient has allowed to use medical records and therapeutic information is attached to the medical document. The authors testify the patient privacy maintenance. 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.The authors declare that there are no conflicts of interest preparing this manuscript and accept any responsibilities.


Primary lymphoma of the gallbladder (GB) is a rare condition, and very few cases have been reported so far. Diagnosis is usually made after surgery of suspicious GB mass, which is often difficult to differentiate from GB carcinoma. The GB wall does not contain lymphoid tissue, and tumors arise at the submucosal layer. Stone disease and chronic inflammation may contribute to its pathogenesis. Treatment consists of surgical resection followed by adjuvant therapy in selected cases. We present a case of an unusual, large-sized mucosa-associated lymphoid tissue (MALT) lymphoma of the GB. Gallbladder (GB) lymphoma accounts for less than 0.2% of all GB malignancies, and very few cases have been reported so far. It can be either primary, in the form of extra-nodal non-Hodgkin lymphoma solely originating in the GB, or secondary (more common), as a result of a general lymphoproliferative disease with or without multiple other sites involved within the gastrointestinal (GI) tract . The mosPathogenesis is not clear as the GB wall lacks lymphoid tissue, but it is hypothesized that chronic irritation and possible bacterial translocation in the background of lithiasis may cause lymphocyte migration and infiltration. In an alternative pathway, an antigenic stimulation causes chromosomal translocation resulting in the synthesis of a fusion protein (API2-MALT1) that inhibits apoptosis and causes antigen-independent proliferation ,5.GB lymphoma has an indolent course and usually does not give systemic symptoms, but most commonly patients report nonspecific upper abdominal symptoms  possibly due to concomitant lithalsas and chronic inflammation. However, stone disease was found only in 65% of cases in one study . The tumWe present a case of a primary GB MALT lymphoma of an unusually large size.A 39-year-old male with a two-month history of low-grade fever, weakness, fatigue, and a weight loss of 5 kilograms was referred to our unit by his general practitioner after he was found to have a palpable right upper quadrant mass. Imaging revealed a 15-cm soft GB mass extending to the central liver parenchyma and hepatic hilum . The most common histological subtypes are MALT, DLBCL, and follicular lymphoma . In one Symptoms are insufficient to direct diagnosis as they are infrequent and non-specific, and if existing, they are usually related to concomitant stone disease . ImagingThe majority of cases reported were discovered incidentally, but if a tumor is suspected on imaging, management should be similar as in GB adenocarcinoma. Open or minimal invasive cholecystectomy with possible GB bed liver resection based on the frozen section of the GB and cystic duct stump is recommended. It has been shown that at least four lymph nodes are required for staging purposes, with six being the number advised by the American Joint Committee on Cancer . InitialDue to the very small number of cases and the fact that confirmation of diagnosis usually follows surgery, a treatment protocol is difficult to establish. Factors that can influence the decision for adjuvant treatment are the presence of distant disease, histological subtype, incomplete surgical margins, secondary GB lymphoma originating from another site within the GI tract, bone marrow biopsy results, and age and fitness of the patient. In general, simple R0 cholecystectomy appears sufficient in the majority of cases. In our case and in the light of a less radical resection, we offered chemotherapy, as described previously. Our patient responded very well and remains free of disease two years after resection.Primary GB MALT lymphomas are particularly rare, and imaging findings are nondiagnostic. Tissue diagnosis is possible with intraoperative frozen section examination. Due to the soft consistency of the tumor, it is possible to achieve a complete resection without necessarily proceeding to liver bed resection at the time of surgery. With the combination of a complete resection and adjuvant chemotherapy, our patient has so far remained free of disease two years after surgery.


Oncocytomas are uncommon benign tumors that arise in various organs and are predominantly composed of oncocytes. Adrenocortical oncocytomas are extremely rare and are generally non-functioning.18F-FDG-PET. Because of the uncertain nature of the growing lesion, it was decided to proceed to surgical resection.We report the case of a 40-year-old patient with a progressively enlarging left adrenal mass. At the age of 19 he had undergone right adrenalectomy for a cortisol-secreting adenoma. Radiologic features were not typical of an adenoma and positive uptake was detected at The surgeon managed to remove the left adrenal mass, sparing the normal adrenal gland, and histology was consistent with adrenocortical oncocytoma. Corticosteroid supplementation was prescribed, but at reassessment, adrenal function was found to be preserved and treatment withdrawn.Adrenal oncocytoma is a rare diagnosis, but should be considered in the presence of a growing mass with non-specific radiologic appearance. Oncocytomas are uncommon benign tumors arising in various organs such as the kidneys, salivary glands, thyroid, parathyroids and pituitary . They arAdrenocortical oncocytomas are exceedingly rare , 5. TheyWe describe the case of a patient with a previous history of cortisol-secreting adrenocortical adenoma, referred to our clinic after the discovery of a contralateral lesion.A 40-year-old man was seen at our clinic in summer 2020 for a left adrenal mass. At the age of 19, he had suffered from ACTH-independent Cushing syndrome, when a right adrenal lesion was detected at CT scan, leading to adrenalectomy. Histopathology was indicative of adrenocortical adenoma. Cortisol normalization occurred after surgery and follow-up visits were scheduled, with regular assessment of adrenal function and abdomen ultrasound imaging. In 2018, a CT scan had revealed a left adrenal mass with a maximum diameter of 3 cm . A diagn18F-FDG-PET scan, with a SUVmax adrenal/SUVmax liver ratio of 3.5 (When we first met the patient, in the summer of 2020, a further MRI indicated disease progression, with the mass measuring 5.5 cm in greatest diameter and presence of inhomogeneities in the T1, T2 and post contrast sequences . The leso of 3.5 . At a muo of 3.5 . Histoloo of 3.5 . The posSince the first description by Kakimoto et al. in 1986 , less thAdrenocortical oncocytic lesions are generally nonfunctional and detected incidentally , as in oAt CT scan or MRI, about 30% of oncocytomas may display a central fibrous scar, resulting in the so-called ‘spoke wheel’ pattern , not obs18F-FDG-PET showed positive uptake, but it should be borne in mind that adrenocortical oncocytomas may display false-positive uptake at 18F-FDG-PET, possibly due to the presence of abundant intracellular mitochondria (18F-FDG into the cells (Oncocytic neoplasms are usually large, surrounded by a fibrous capsule, and exhibit a non-invasive growth pattern . Ultrasohe cells , 20.On account of the increase in size and uncertainty in radiologic characterization of the lesion, it was decided to proceed with surgical removal. In principle, total adrenalectomy would have been the treatment of choice, in view of the uncertain diagnosis. The patient was informed of the risk of adrenal insufficiency following surgery, in view of previous right adrenalectomy. However, the fact that a clear cleavage plane between the lesion and the adrenal gland had been identified, allowed the selective removal of the tumor, while sparing the normal gland.Pathologic examination revealed the lesion to be an oncocytoma. As reported in the literature, diagnosis of oncocytoma is based on morphologic and immunohistochemical features. Electron microscopy demonstrates numerous mitochondria in the tumor cell cytoplasm . At immuIn 2004, Bisceglia et al.  modifiedA unique characteristic of our case was that right adrenalectomy had previously been performed for Cushing syndrome due to a cortisol-secreting adenoma. Although the left adrenal gland was spared at surgery, a strict monitoring was carried out over subsequent days. While blood pressure, glycemia and serum electrolytes showed no alterations, the patient was discharged with caution and prescribed corticosteroid supplementation. Adrenocortical function was reassessed after two months and found to be normal. Cortone acetate supplementation was then discontinued, and the patient is regularly followed up at our outpatient clinic.In summary, we have described here a rare case of adrenocortical oncocytoma in a patient previously subjected to contralateral adrenalectomy for Cushing syndrome due to a cortisol-secreting adenoma. The presence of a growing adrenal mass of uncertain origin in a patient with only one adrenal gland, to our knowledge an unprecedented condition, offered a clinical challenge calling for a multidisciplinary approach. Although adrenal oncocytoma is a rare diagnosis, it should be considered in the presence of a well-demarcated, enlarging mass with non-specific radiographic characteristics.


Congenital (or primitive) cholesteatoma is a benign disease with slow progressive growth that destroys neighboring structures. It is considered an epidermal cyst originating from the remnants of squamous keratinized epithelium. The disease may appear in several regions of the temporal bone such as in the middle ear (most frequent site) as well as in the petrous apex, cerebellopontine cistern, external acoustic meatus and mastoid process. Congenital cholesteatoma of the mastoid process is the rarest form of presentation in the temporal bone.The aim of this report was to present clinical and imaging features of a patient with an isolated congenital cholesteatoma of the mastoid process, and to provide a brief review of the literature.tinnitus in the left ear. She had no history of ear infections or surgery. Physical examination revealed mild pain upon compression of the left mastoid process, and occasional transient dizziness. Both tympanic membranes were normal. Tuning fork and Weber tests were unremarkable. Audiometric evaluation demonstrated bilateral sensorineural hearing loss, particularly for high frequency sounds, with good discrimination. Tympanometry and stapedial reflexes were normal. Brainstem electric response audiometry (BERA) also presented normal results. Computed tomography (CT) scan of the temporal bones showed a hypoattenuating and expansive lesion in the left mastoid process, eroding its medial and lateral walls adjacent to the left sigmoid sinus (tinnitus. Follow-up CT scan confirmed no recurrence and showed a large surgical cavity in the left mastoid process communicating with the antrum, aditus ad antrum, and tympanic cavity. Bone erosion occurred adjacent to the left sigmoid sinus (A 67-year-old black female from Santos city, São Paulo State, Brazil, had sought medical assistance 5 years earlier for bilateral hearing loss and an 8-year history of pulsatile id sinus . The remid sinus .Figure 1tinnutus may have been due to the lesion's contiguity with the adjacent non-thrombosed left sigmoid sinus. The differential diagnosis of this lesion can be reached based on a finding of cholesterol granuloma. However, cholesterol granulomas are typically hyperintense on T1-weighted images and other rare manifestations include endolymphatic sac tumor and meningiomas.In 1953, HouseWe described a patient with a rare isolated congenital cholesteatoma of the mastoid process that presented typical clinical and imaging features. The condition, although extremely rare, may be suspected based on clinical and imaging characteristics. However, this suspicion requires subsequent histological confirmation. Management is by surgical treatment with full lesion resection and periodic postoperative follow-up.


Gallstones are the most common cause of acute pancreatitis (AP). Walled-off pancreatic necrosis (WOPN) is one of the sequelae of AP. Endoscopic and laparoscopic techniques for cystogastrostomy have been reported in the literature as treatment options for complicated or symptomatic WOPN. Here, we describe two cases of gallstone-related AP complicated by WOPN treated by robotic-assisted transgastric cystogastrostomy and cholecystectomy. Acute pancreatitis (AP) is an acute inflammatory condition of the pancreas. Gallstones are the most common cause of AP . Other cSevere AP comprises about 25% of all pancreatitis patients. It is a life-threatening condition with an in-hospital mortality rate of around 15% . A more Acute necrotic collection is a peripancreatic collection associated with pancreatic necrosis. It consists of fluid and necrotic tissue (of the pancreatic parenchyma and/or peripancreatic tissues). After weeks, it is usually called walled-off pancreatic necrosis (WOPN). WOPN is an encapsulated collection formed of pancreatic and/or peripancreatic necrosis with a well-defined, mature, inflammatory wall. Maturation usually occurs at least four weeks from the onset of AP ,11.Here, we present the cases of two patients who underwent robotic-assisted cystogastrostomy and pancreatic debridement for WOPN. The first patient was a 52-year-old female patient who presented with epigastric pain radiating to the back, in addition to intolerance for oral intake for a month. She had a medical history of chronic liver disease secondary to hepatitis C virus infection. She underwent three cesarean sections. Five weeks before the presentation, she was admitted to another hospital with a clinical picture suggestive of acute gallstone pancreatitis and acute cholecystitis, which was managed conservatively. Her condition improved, and she was discharged two weeks later; however, vomiting, abdominal pain, and intolerance for oral intake continued. She lost 15 kg weight over a month.2. She was incapable of lying flat due to the pain. Her abdominal examination revealed epigastric fullness and tenderness. Although there was a mild elevation in serum amylase and lipase levels, she had normal laboratory results. Abdominopelvic computed tomography (CT) scan with contrast  was 38.3 kg/mt Figure  showed n2. He reported episodes of vomiting after meals which made him refuse oral intake. Apart from mild anemia and hypoalbuminemia, his lab results were within normal limits. Abdominopelvic CT scan showed two large collections, and the gallbladder was harboring multiple stones with a thick edematous wall, no intra or extrahepatic biliary radical dilatation, and a CBD stent in place. The first collection was in the lesser sac with a mature wall, and the second was an encapsulated subhepatic collection (related to the left lobe). We decided to insert an ultrasound-guided percutaneous draining catheter in the subhepatic collection a couple of days before surgery.The second patient was a 31-year-old male. He presented with abdominal pain and swelling with a history of acute severe pancreatitis post-endoscopic retrograde cholangiopancreatography (ERCP) for extraction of a common bile duct (CBD) stone. On admission, the patient had features of acute weight loss, with an obvious abdominal mass. His BMI was 20.5 kg/mBoth patients were scheduled for robotic-assisted laparoscopic cystogastrostomy and cholecystectomy. The surgical procedures were essentially the same for both patients. Patients were placed in the supine position and received general anesthesia. The open technique was used to insert the scope port. After acquiring sufficient pneumoperitoneum, the working ports were inserted under vision, two ports for the robotic instruments, and two for assistance. On exploration of the abdominal cavity, adhesions were noted and sharply cut. The subhepatic collection of the second patient was deroofed and cleaned. The stomach was identified as displaced anteriorly by the WOPN. An anterior gastrotomy was done Figure . The posAfter irrigation and suction, the posterior gastric wall was sutured together with the WOPN using running 3-0 polydioxanone (PDS) sutures. For each patient, a nasogastric tube was placed in the stomach under vision. The anterior gastrotomy was also sutured using running 3-0 PDS sutures and an omental patch was placed.Cholecystectomy was then started by obtaining the critical view of safety. The cystic duct and artery were identified, secured with metallic clips, and divided. Two intra-abdominal drains were placed in each case. Surgery time was around five hours in the first case and four hours in the second case. The blood loss was about 150 mL in the first case and 200 mL in the second case.The first patient was transferred to the intensive care unit for a single day. She had an uncomplicated postoperative course apart from a single attack of fever of 38°C on the first postoperative day. She started oral intake on the fourth postoperative day. She was discharged on the sixth postoperative day. The patient’s symptoms dramatically improved. On the first postoperative day, her epigastric pain disappeared, and she was able to lie on her back without pain. On follow-up visits, the patient had no pain, and she regained tolerance for food with no nausea or vomiting.The second patient was transferred directly from the operation theater to the ward. He started oral fluids on the third postoperative day and was discharged on the seventh postoperative day. All his symptoms improved, and he started to gain weight in the next follow-up visits. A month later, he suffered from epigastric pain and vomiting, which were controlled with a short course of proton-pump inhibitors and prokinetics.This report presents two cases with WOPN, post-gallbladder stones related AP, treated with a robotic-assisted laparoscopic procedure.For years, open surgical pancreatic debridement or necrosectomy was the standard of care for patients with WOPN or necrotizing pancreatitis . HoweverRobotic cystogastrostomy was also reported for the management of WOPN in case reports ,16 or smThe overall experience with the robotic-assisted transgastric cystogastrostomy for the two cases, with relatively large WOPN, and one of these cases was morbidly obese, was very satisfactory. Even though they were chronologically considered to be the early cases done in our young robotic surgery program, the Ain Shams University Specialized Hospital robotic program only started on October 31, 2021. It is worth mentioning that, on reviewing reports from the manufacturing company of the robotic system used in those cases and by reviewing literature from Egypt, we did not find any previous similar cases. This makes these two procedures the first robotic-assisted cystogastrostomy performed with this platform worldwide and the first to be done in Egypt.Robotic-assisted transgastric cystogastrostomy with pancreatic debridement and cholecystectomy for patients suffering from WOPN and gallbladder stones is a safe and feasible procedure, even with large cyst volumes. The potential benefits of the robotic approach over the conventional laparoscopic approach may be outweighed by the relatively higher cost. Comparative studies are needed to answer this question.


Insulinoma is an insulin-secreting tumor that causes hypoglycemia due to inappropriately high insulin secretion. The Whipple's triad, which comprises indications of hypoglycemia , plasma glucose concentration <55 mg/dL (3.0 mmol/L), and resolution of symptoms after administration of glucose, is utilized for the determination of insulinoma. In this report, we present the case of a thin, lean, adult male with a BMI of 22, who presented with repetitive episodes of tremor, sweating, weariness, and perplexity that occurred amid fasting and settled with meals, fulfilling Whipple's triad criteria for the determination of insulinoma. The episodes frequently led to seizures. Supervised fasting was carried out, which revealed raised C-peptide levels, low blood glucose, and negative sulfonylurea screen. A computed tomography (CT) scan localized the tumor, and surgical resection was planned. Insulinoma is a small insulin-secreting tumor that causes hypoglycemia due to inappropriately high insulin secretion. It is a type of functional neuroendocrine tumor (NET) and is exceptionally uncommon with a yearly frequency of 1 to 4 per million per year . We presThe therapy of choice is surgical resection. Imaging studies, including a CT scan, localize the tumor before resection. The enucleation of insulinoma and partial distal pancreatectomy are the two most widely employed methods . Other cFor the past three years, a 20-year-old slender, lean adult male complained of increased hunger, tremors, perspiration, palpitation, confusion, and frequent seizures. Within the last four months, these symptoms got worse. According to the attendant, incidents usually happened late at night or in the early hours of the morning while he was fasting, and they lasted anywhere from a few minutes to several hours. The patient denied having any visual field issues or headaches and said he had never abused drugs. His past medical and surgical records were unremarkable.A young patient lying quietly in bed without distress on general physical examination was found to have scanning speech and horizontal nystagmus, with the rest of the systemic examination results being normal.High level of blood insulin  and low level of blood glucose  were revealed by initial laboratory tests. Other important profiles such as renal, thyroid, hepatic, coagulation, bone, prolactin, and parathyroid were all in the normal range, along with complete blood count. The patient was hospitalized for additional testing due to recurrent hypoglycemia symptoms and a potential pancreatic insulinoma. During a hypoglycemic episode  that occurred during prolonged (48-hour) fasting, his insulin and C-peptide levels were examined. Laboratory findings during the event revealed low blood glucose (37 mg/dL), increased blood insulin (190 pmol/L), and high C-peptide  values. Sulfonylurea or meglitinide toxicity was not seen in the blood. Dextrose injection significantly alleviated hypoglycemia symptoms, and Whipple's triad for insulinoma was satisfied.An abdominal CT scan revealed a rounded arterialized lesion measuring 12x10 mm in the posterior aspect of the pancreas’ neck  was excluded based on history, physical examination, and laboratory Investigations . Serum prolactin was 11 ng/mL, PTH was 23 pg/mL, and serum calcium 9.2 was mg/dL. All these normal investigations rule out the diagnosis of MEN1.The patient was referred from our medical unit to a laparoscopic surgeon for tumor removal via Whipple's procedure.An insulinoma is a neurosecretory tumor that results in the overproduction of insulin, leading to a state of hypoglycemia. The majority of insulinomas are benign and of solitary character. Due to the "rule of 10," i.e., 10% demonstrate multiplicity, 10% are connected with MEN1, 10% are malignant, and 10% are ectopic . The sigThe diagnosis of insulinoma is on the basis of the Whipple triad, which includes symptoms of neuroglycopenia  and sympathoadrenal symptoms  in the existence of low serum glucose level (<50 mg/dL) that resolves following the administration of glucose .The prevalence of insulinoma is reported to be about 1 to 4 per million per year . The sigA range of techniques is applied to localize an insulinoma, which includes transhepatic portal venous sampling, arteriography, transabdominal ultrasonography, intraoperative ultrasonography, abdominal computed tomography (CT) scan, magnetic resonance imaging (MRI), transhepatic portal venous sampling, endoscopic ultrasonography (EUS), intraoperative palpation, selective arterial calcium stimulation with hepatic venous sampling, and 111 In-labeled octreotide scans with single-photon emission tomography. The sensitivity of some of the methods such as MRI, CT scan, and EUS reported is 37-71%, 30-66%, and 37-71%, respectively, for pancreatic insulinoma identification, whereas the combination of MRI, CT scan, and EUS has reported to identify insulinoma is 17-66%. In addition, arteriography was regarded the gold standard for insulinoma localization. However, documented sensitivities of 29-64%, paired with superior noninvasive imaging techniques, have restricted its use .Overall, the survival of people having insulinoma is high in the long term, and it is estimated that roughly 90-95% of insulinomas have benign histological behavior; therefore, resolution with the absence of symptomatology after full excision is the rule . Among 1In malignant insulinomas, the long-term survival is imaginable, mainly in instances firmly limited to the pancreas and with strong response to adjuvant chemotherapy; median disease-free survival after curative excision is estimated at five years. The median survival time observed after the recurrence of illness is 19 months .Insulinoma is a sporadic tumor that is more often found in obese patients. Due to increased appetite and repeated episodes of hypoglycemia, patients ordinarily have hyperphagia, leading to obesity. However, our patient with insulinoma is uncharacteristically lean with a BMI of 22. In this way, doctors ought to have a large index of suspicion in patients presenting with repetitive hypoglycemic events that resolve with meals.


Epidermoid cysts (EC) are benign lesions resulting from incomplete separation of the neuroectoderm during embryonic development. The investigation of choice for EC is magnetic resonance imaging (MRI). Surgical resection is the treatment of choice. Full resection of EC including the cyst wall to prevent recurrence and malignant transformation should be considered when possible. Two main approaches were described in the literature and included craniotomy and endoscopic endonasal approach (EEA). Using of EEA to accomplish total resection could be challengeable. To best of our knowledge, only 6 manuscripts  reported total resection of EC by using EEA. Our case should be the ninth such cases in the literature. In this paper, we reported a case of sellar and suprasellar epidermoid cyst which was resected completely by using EEA. We revealed the safety and efficacy of this approach in management of such cases. Intracranial epidermoid cysts (EC) are rare, congenital and benign cystic lesions. They account for 0.04%–1% of intracranial tumors. The lesions often become symptomatic in third and fourth decade of life . They arA 22-year-old caucasian male was admitted to neurosurgery department with a history of chronic headache and 8-month history of bilateral visual acuity deterioration. Ophthalmologic evaluation revealed light perception on the right side and hand motion on the left side. Fundus examination showed bilateral temporal optic nerve pallor. Eye movement was normal. The patient had no focal neurological deficits. Laboratory tests were unremarkable. Magnetic resonance imaging (MRI) revealed a sellar and suprasellar well-defined cystic lesion with irregular margins, measures about 2.6 × 3.3 × 4 cm. The mass was hypointense in T1-weighted images (T1WI) and hyperintense in T2-weighted images (T2WI) with restriction in diffusion-weighted images (DWI). The mass was compressing the optic chiasm and surrounding the right optic nerve and pituitary stalk . The pat. In our case, we successfully achieved complete resection by using endoscopic surgery and transnasal trans-sphenoidal approach. The patient had postoperative aseptic meningitis which was treated conservatively. It is important to keep in mind chemical meningitis due to intrathecal spillage of the cyst content during surgery [An intracranial EC is a congenital lesion results from trapping ectodermal remnants inside the cranium during embryonic development . The slo surgery . In the  surgery .


Lipomas are one of the commonest soft tissue tumors which usually arise from subcutaneous plane. Lipomas can progress to one of the three types of liposarcomas. Dedifferentiated liposarcomas have metastasis potential, risk of recurrence and increased risk of mortality. The culture of taking every lipoma as benign should be seriously considered with different approaches as it can lead to sarcomatous degeneration in some cases.A 78 years old male patient present to our hospital with recurrent left thigh swelling of two years duration with limping and sense of heaviness on left side. He has history of previous surgery for the same complaint on the same site two times, six and four years ago. Histological diagnosis in both cases was lipoma. Objectively he has 20 × 60 cm non tender, soft to firm, slightly mobile swelling on left thigh spanning anterior, lateral and posterior compartment but no inguinal swelling. Fine needle aspiration cytology from the mass reported sarcoma. CT-scan stated diagnosis as sarcomatous tumor.Lipoma is commonest soft tissue tumor arising from adipocytes more often in young adults, women and overweight population. Well-differentiated liposarcoma can progress to dedifferentiated liposarcoma over time which is slightly aggressive than its precursor. Management of liposarcoma depends on stage, site and histology. Surgical resection with radiotherapy is the standard of care for localized dedifferentiated liposarcomas.It is difficult to differentiate between giant lipoma and well-differentiated liposarcoma with physical examination and imaging. Histopathologic examination in uncertain conditions will decrease morbidity and mortality from the more aggressive forms of liposarcomas. •Lipoma is one of the commonest soft tissue tumors which usually arise from subcutaneous plane.•Well- differentiated liposarcomas more resembles lipoma except for its enlarged nuclei and some degree of atypia.•Dedifferentiated liposarcoma has metastasis potential, risk of recurrence and increased risk of mortality.•Surgical resection with radiotherapy is the standard of care for localized dedifferentiated liposarcomas. As it arises from fat cells, it can be found in any plane/part in the body Three main types of liposarcoma include well-differentiated/dedifferentiated, myxoid/round cell and pleomorphic. Well-differentiated and dedifferentiated liposarcomas are most common types and share the same genetic alteration 2A 78 years old male patient presents to our hospital with recurrent left thigh swelling of two years duration with limping and sense of heaviness on left side. He has history of previous surgery for the same complaint on the same site two times, six and four years ago. The diagnosis which was confirmed histologically after both resections was lipoma. There was no further treatment after resection. The patient has no other site swelling, cough, bone pain or abdominal pain. The patient has no other history of chronic illness.Objectively, he was comfortable with stable vital signs. There was no lymphadenopathy, chest was clear and resonant. There are two previous incision sites on left thigh. There was 20 × 60 cm non tender, soft to firm, slightly mobile swelling on left thigh spanning anterior, lateral and posterior compartment, no inguinal swelling. All blood work (complete blood count and organ function tests) were normal. Fine needle aspiration cytology from mass reported spindle cell tumor with mixed mesenchymal and fatty cells. Chest X-ray and abdominal ultrasound for metastasis were negative. CT-scan of chest, abdomen and pelvis for metastasis were free. CT-scan of the involved extremity showed 20 × 50 × 20 cm left thigh soft tissue mass involving anterior, lateral and posterior compartment extending from supra-knee area to mid of femoral triangle and up to gluteal region (10 cm below iliac crest) postero-laterally. There is no involvement of femoral vessels and there is no inguinal mass. Fine needle aspiration cytology from the mass reported sarcoma. CT-scan stated diagnosis as sarcomatous tumor A-D.Fig. Patient was prepared for operation with informed consent and prophylaxis antibiotics. We made Lazy-S incision over the mass. There was 20 × 60 cm mass soft to firm consistency, mostly of fatty appearance and firm on superior aspect. The mass adheres to femoral sheath and infiltrates some part of sartorius and vastus lateralis muscles but no neurovascular invasion A-C. Wide3Lipoma is commonest soft tissue tumor arising from adipocytes more often in young adults, women and overweight population. When the size enlarges to more than 5 cm, it alerts other malignant lipomatous tumor transformation Well-differentiated liposarcoma can progress to dedifferentiated liposarcoma over time which is slightly aggressive than its precursor. In a study by Thirasastr and Somaiah, it was found that 10 % of liposarcoma arise as recurrence after resection of well-differentiated liposarcoma, while the rest 90 % progressed from undiagnosed well-differentiated liposarcoma Management of liposarcoma depends on stage, site and histology. Surgical resection with radiotherapy is the standard of care for localized dedifferentiated liposarcomas. Multimodality approach for advanced diseases is amenable although prognosis is poor in this group of patients abemaciclib and the nuclear export inhibitor SelinexorManagement of advanced liposarcomas consists of systemic chemotherapy, radiotherapy and surgical resection whenever possible. Systemic therapies historically limited only to cytotoxic chemotherapeutic agents. Recent studies showed improved effects with combination of cytotoxic agents and tyrosine kinase inhibitors. Clinical trials showed target therapies with cyclin-dependent kinase 4 (CDK4) inhibitor It is difficult to differentiate between giant lipoma and well-differentiated liposarcoma with physical examination and imaging. Histopathologic examination in uncertain conditions and the above precursor cases will decrease morbidity and mortality from the more aggressive forms of liposarcomas.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-review.This work does not receive funds.Not applicable.Nebiyou Simegnew contributed substantially from the patient evaluation to writing up, Amare Amare contributed in revision of the paper.1.Name of the registry: researchregistry.com.2.Unique identifying number or registration ID: researchregistry7735.3.https://www.researchregistry.com/browse-the-registry#home/.Hyperlink to your specific registration (must be publicly accessible and will be checked): Nebiyou Simegnew will take the primary responsibility of the study.The authors declare that they have no competing interests.


Case Description. 37-year-old female presented with a 1-day history of abdominal pain. CT abdomen identified a 12.5 cm heterogeneously enhancing left adrenal mass. Plasma/urine catecholamine and metanephrine levels were markedly elevated with evidence of elevated serum/urine cortisol. However, the patient's subsequent urine toxicology was found to be positive for amphetamines, which she later admitted to using, 1 week prior to admission. Repeat biochemical workup after 1 week drug washout period showed improvement in both catecholamine and cortisol levels. Given the high degree of suspicion for PCC, an open laparoscopic adrenalectomy was performed with histology confirming SDHB gene mutation positive giant pheochromocytoma. Discussion. Diagnosis of PCC in a patient with a history of amphetamine abuse remains an enigma, to which addition of it being a giant PCC that are rare and typically silent further confounds the clinical picture as seen in this case.  Diagnosis of giant pheochromocytoma is difficult; patients often lack the classic triad and presence of gross biochemical positivity. At times, presence of sympathetic stimulant drugs can further complicate the clinical picture. Here, we present a case of giant “functional” pheochromocytoma with a history of amphetamine use.  PCC could be termed a “chameleon” tumor given its varied clinical presentations and lack of standardized biochemical and radiological data . Pheochromocytomas (PCCs) are rare catecholamine producing tumors with lethal effects if not diagnosed and treated in a timely manner. On the other hand, surgically treated PCCs have an almost 90% 5-year and 78.9% 10-year survivability rate, as reported in a large Denmark-based study [The biggest limitation in PCC management remains its diagnoses as classic triad are often paroxysmal  and hypeGiant pheochromocytomas are categorized as tumors >7 cm in size , 7. ThesA 37-year-old female with a past medical history of polysubstance abuse and generalized anxiety disorder (GAD) presented to ED for sudden onset of abdomen pain for 1 day. CT abdomen/pelvis with contrast showed a 12.5 cm heterogenous enhancing left adrenal mass with mild to moderate hyperdense fluid throughout the abdomen from a presumed previous hemorrhage . The patThe patient denied any history of hypertension, diaphoresis, but did report occasional headaches, palpitations, and a 10–15 lbs weight loss over the last 4 months. She denied any current substance use. Medication history included suboxone and Wellbutrin. Family history was otherwise negative for any endocrine disorders.2 sat of 99% on room air. On physical examination, she was found to have abdominal distention and diffuse abdominal pain with diffuse anasarca. A focused exam was negative for any cushingoid features. The biochemical workup revealed plasma cortisol 6 times ULN, plasma metanepherines 25–30 times ULN, and urine metanepherines 30–100 times ULN. Detailed lab values and normal reference ranges for our institution are reported in Hemodynamically she had mild tachycardia with HR 112 bpm, BP 114/92 mmHg, temp 98.6°C, respiratory rate of 18 breaths per min, and an OGiven the still posed diagnostic dilemma, an MIBG scan was ordered, however due to institutional limitations, could not be conducted. Differential diagnosis at this point, in order of most to least likely, included giant PCC, large corticomedullary tumor, and nonfunctioning adrenal adenoma with substance induced hypercortisolemia/catecholamine surge.The decision regarding optimal preoperative management was complicated by an unclear tumor type. However, given higher probability of PCC, she was started on 1 mg doxazosin twice daily with a maximum tolerated dose of 6 mg twice daily, 12.5 mg metoprolol twice daily with a max dose of 25 mg twice daily. This did cause the patient's BP to drop, systolic BP in 80s, which was treated with aggressive 1-2 L intravenous fluid hydration (IVF) and 1-2 g sodium tablets per day. She then started to third space likely due to poor albumin reserve related to malnourishment , requiring albumin infusion.An open laparotomy was performed which showed a rock-hard mass with associated inflammation and hemorrhaging involving the adrenals, spleen, and tail of pancreas. A left adrenalectomy, splenectomy, and distal pancreatectomy was performed. The patient's intraoperative and postoperative vitals remained stable and she was safely titrated off both beta and alpha blockers day two postoperation. The surgical pathology identified a 10.5 cm left giant pheochromocytoma with a mixed hemorrhagic and cystic center. It was well encapsulated within the fibrous. No lymphovascular or fat invasion was observed. Immunohistochemical staining was positive for chromogranin A and INSM1 with loss of SDHB expression in tumor cells. No metastasis was identified in pancreatic or splenic tissue. Patient's plasma metanephrines improved to normal by day 3 .She is now being monitored periodically for recurrence of disease with surveillance biochemical workup and CT abdomen/pelvis due to positive SDHB mutation.Catecholamines such as epinephrine, dopamine, and norepinephrine are released by chromaffin cells within the adrenal medulla. An excess presence of these cause symptoms of headaches, palpitations, and diaphoresis , 4. ThisMedications such as SSRI/SNRI's and amphetamines can increase extracellular catecholamine levels , 9. AmphGiven posed diagnostic dilemma, imaging modalities are often used next to improve the diagnostic sensitivity. However, even on imaging, PCCs may have atypical presentations as fatty, cystic, or calcified masses with variable signaling on T2 MRI and variable degree of contrast washout on CT . FunctioOne could assume that larger PCCs would be associated with higher catecholamine release and thus present with florid symptoms associated with catecholamine surge making diagnosis of PCC easier. On the contrary, to the best of our knowledge all giant pheochromocytomas reported in the literature thus far have been silent with mild catecholamine elevation often presenting as incidental findings on imaging or with abdominal pain due to mass effect –16. The We presented here a unique case of a giant 10.5 cm “functional” pheochromocytoma with several diagnostic dilemmas posed by atypical presentation and amphetamine abuse history. Although initially seen grossly positive urine/plasma catecholamines and metanephrines levels made diagnosis of PCC a high probability, presence of concomitant hypercortisolemia and >50% decline in catecholamines and cortisol levels after 1 week of drug washout period added further complexities around diagnosis. The diagnosis of PCC was confirmed after the tumor resection and improvements in her catecholamine levels after tumor resection .Giant PCCs are rare entity in itself but those reported are typically silent, thereby making our case the first one to report a functional giant PCC. Diagnosis of “functional” giant PCC would have otherwise been straight forward had it not been for our patient's use of amphetamines.