Pituitary adenomas are one of the most common intracranial tumors. Non-functioning macroadenomas are usually diagnosed when they cause symptoms due to the mass effect on surrounding structures. We present the case of a 48-year-old man who presented with a headache associated with ptosis of the right eye and right-sided blurry vision for three days. Initial computerized tomography (CT) scan of the head did not report a mass, acute infarct, or hemorrhage. He was given 325mg of aspirin for concern of a stroke while waiting for magnetic resonance imaging (MRI) of the brain, which was done the next day and revealed a pituitary macroadenoma with hemorrhage, mass effect, and compression of the optic chiasm consistent with pituitary apoplexy. He ultimately underwent trans-sphenoidal resection of the tumor; however, his surgery was delayed for five days as he had received a high dose of aspirin in the Emergency Room. His adrenocorticotropic hormone (ACTH), cortisol, and testosterone levels were found to be quite low. He was administered stress dose steroids peri-operatively and ultimately discharged on indefinite hydrocortisone therapy and endocrinology follow-up. Our case highlights a serious complication of pituitary adenomas that can occur called pituitary apoplexy which is caused by acute ischemic infarction or hemorrhage in the pituitary. It needs prompt identification and management. Our case also emphasizes the importance of recognizing pituitary apoplexy as one of the causes of sudden onset cranial nerve deficits, as it is a rare presentation of pituitary adenomas that can be taken for a stroke in the Emergency Department. Pituitary adenomas are the fourth most common intracranial tumors as per the American Association of Neurological Surgeons . Most ofA 48-year-old man with a history of cirrhosis, diabetes, hypertension, and end-stage renal disease (ESRD) presented to the Emergency Room for evaluation of a headache, associated with ptosis of the right eye and right-sided blurry vision. Symptoms had started three days prior to presentation and progressively worsened, prompting him to come to the hospital. The headache was sudden in onset, temporal in location, radiating to the frontal region, and progressive in severity. He rated it as 10/10 in intensity at the time of presentation. He did not report any dizziness, numbness, or focal weakness anywhere else in the body. On exam, he was alert and oriented with normal speech. He was mildly hypertensive with a blood pressure of 152/77 mm Hg. The rest of his vitals were within normal range. He was noted to have complete ptosis of the right eye along with medial gaze palsy of the right eye. Pupils were constricted bilaterally with a sluggish response to light. No anisocoria was noted. The fundoscopic examination was limited due to pupillary constriction. Visual fields were noted to be intact to confrontation, and facial movements were symmetric. The rest of his exam was unremarkable. His initial labs were within the expected range of the intradialytic period. CT scan of the head on admission reported no intracranial hemorrhage, infarction, or mass. A CT Angiogram of the head and neck was unremarkable. He was given aspirin 325mg for concern of stroke, and an MRI of the brain was ordered, which was not done until the next day. Meanwhile, he was given metoclopramide, diphenhydramine, and ketorolac for headaches with minimal relief. The findings on the MRI of the brain came as a surprise but explained the patient’s symptoms very well. It revealed a 2.1x2.1x1.4 cm rim-enhancing sellar/suprasellar mass causing a mass effect on the optic chiasm. This was followed by a dedicated pituitary fossa MRI which confirmed enlargement of the pituitary gland consistent with macroadenoma with suprasellar extension and hemorrhage in the lesion, causing optic chiasm compression and displacement upwards. He had developed pituitary apoplexy that likely occurred several days ago. Neurosurgery was consulted immediately, and the patient was scheduled to undergo trans-sphenoidal resection of the tumor; however, unfortunately, surgery had to be delayed for five days as he had received aspirin 325mg on admission. Otolaryngology was consulted for co-management of this procedure and endocrinology was consulted for possible complications after surgery. Hormone levels were checked, and he was found to have low levels of adrenocorticotropic hormone (ACTH) and cortisol, consistent with central adrenal insufficiency, hence, he was started on hydrocortisone replacement therapy. He was also found to have other hormonal abnormalities which are shown in Table This case highlights the importance of broadening our differentials and including pituitary tumors as a possible etiology when patients present with cranial nerve deficits. It also demonstrates a level of anchoring bias due to which this case was initially diagnosed as a stroke and treated with a loading dose of aspirin, delaying his surgery.What makes our case more interesting is the fact that the pituitary adenoma was not picked up on the initial CT scan of the head, which is unusual given its size. A subsequent review of the initial CT imaging by a neuroradiologist later stated that although it was present on the initial CT scan but was difficult to see due to streak artifact. Hence, this case also emphasizes keeping in mind the inter-observer variability of interpretations and limitations of imaging modalities. We should not over-rely on imaging reports and should always question them when they don’t correlate clinically.Pituitary apoplexy is a relatively rare but serious complication of pituitary adenomas which occurs in 2%-12% of the cases as mentioned by Briet et al. . It occuThe main symptom of pituitary apoplexy is a sudden onset headache which is severe in intensity. Other symptoms may include visual disturbances such as blurry vision, diplopia, visual field cuts, and ptosis. Sani et al. report an unusual case of pituitary apoplexy in the British Journal of Medical Practitioners, 2020  in whichCT scan (most of the time) and MRI of the brain are diagnostic as they reveal a pituitary tumor associated with hemorrhage. Prompt diagnosis and management are essential to prevent adverse outcomes, as most of the patients develop pituitary insufficiency as a consequence. Once the diagnosis is made, a multidisciplinary team including a neurosurgeon and an endocrinologist should be consulted to co-ordinate an appropriate management plan as outlined by Rajasekaran et al. . The typCentral diabetes insipidus (CDI) is a common complication that can occur after pituitary surgery, whereby kidneys excrete an abnormally large volume of dilute urine, resulting in high serum osmolality. Factors that increase the risk of this complication include large tumor size, gross total resection, and intra-operative cerebrospinal fluid leak among others. It is important to recognize and manage CDI in time as it can cause high morbidity and mortality . Post-suIn conclusion, we present the case of a 48-year-old man who presented with headache and unilateral third nerve palsy and was ultimately found to have a pituitary macroadenoma associated with hemorrhage in the pituitary consistent with pituitary apoplexy. He was noted to have compression and displacement of the optic chiasm. He developed adrenal insufficiency as a consequence and was started on hydrocortisone replacement therapy. He ultimately underwent a trans-sphenoidal resection of the pituitary tumor and was discharged with endocrinology and neurosurgery follow-up. Pituitary apoplexy is a serious complication of pituitary adenomas and early diagnosis, and management is essential to improve outcomes.


Dear Editor,The relationship between the extent of glioma resection and the actual clinical benefit of patients depends on the balance between cytoreductive surgery and neurological morbidity.On May 6, 2022, a 48‐year‐old male suffered a sudden loss of consciousness with convulsions and incontinence that lasted about 10 min. A craniocerebral MRI was performed after he was admitted to the hospital. A space‐occupying lesion, possibly a glioma, was discovered in the right frontal lobe of the brain, with T2 and Flair high signals and no significant enhancement, resulting in an epileptic seizure Figure . On May We selected typical tissues from the same site to be submitted for intraoperative frozen histopathology and rapid molecular diagnosis, respectively. Resect the lesion along the periphery based on experience. About 30 min later, a low‐grade glioma was considered by rapid frozen histopathology examination. Another 25 min later, the isocitrate dehydrogenase 1 (IDH1) R132H mutation and telomerase reverse transcriptase promoter (TERTp) C250T mutation were indicated by rapid molecular diagnosis Figure . Based oThe success of oncological surgery is increasingly defined by the extent of resection, with the goal of complete removal or maximum safe cell reduction. Numerous studies have shown that maximum safe resection has significant prognostic benefits through improved overall survival (OS) and has become the basis for multidisciplinary treatment of lower and higher‐grade brain tumors. Although multiple surgical guidance and imaging modalities exist, iMRI provides the highest evaluation of image quality for surgical implementation.An increasing number of molecular markers have been proven to play an essential role in the classification, typing, grading, prognosis, and treatment of gliomas.The authors have no personal, financial, or institutional interest in any of the drugs, materials, or devices described in this article.Appendix S1Click here for additional data file.Video S1Click here for additional data file.


Rupture of an abdominal aortic aneurysm (AAA) is a surgical emergency with significant associated mortality. This case report discusses a 70-year-old woman presenting to the emergency department (ED) with vague and progressive abdominal pain and relatively benign vital signs and physical exam. She was ultimately diagnosed with a ruptured infrarenal 6.8cm abdominal aortic aneurysm seen on computerized tomography (CT) imaging. Found to be stable, she was transferred to an affiliate tertiary care facility and underwent endovascular repair of the aneurysm. The surgery was successful, and she has since recovered nicely. As reported at her most recent follow up appointments. A timely diagnosis of a ruptured AAA is vital for any chance of a successful outcome. However, in patients with normal vital signs and an unremarkable physical exam, this diagnosis can be incredibly difficult. As such, the need for a broad differential in the setting of abdominal pain is imperative to avoid missing the diagnosis. The use of point of care ultrasound (POCUS) can further aid in this process by providing a quick and affordable screening option to rule out potential aneurysms.Abdominal Aortic Aneurysm, Ruptured Abdominal Aortic Aneurysm, CT imaging, Tangential Calcium Sign, Point of Care Ultrasound. The diagnostic assessment of this patient’s presentation was challenging. Her mild tachycardia and relative hypotension did not depict an individual with hemodynamic instability expected in the case of a ruptured AAA. Once the diagnosis was made, vascular surgery at an affiliate tertiary care center was consulted, and the patient was immediately transferred. The patient underwent emergent endovascular repair of her ruptured aneurysm and was discharged a few days later. At her one-month follow-up, she was asymptomatic and progressing well.The associated images demonstrate the transverse, sagittal, and coronal views of a 6.8 cm infrarenal ruptured AAA continuous with a 4 cm right common iliac aneurysm . Active hemorrhage was seen contained within the aortic wall, and retroperitoneal bleeding can be appreciated with asymmetric enlargement of the left psoas muscle .2Risk of developing an AAA has been increasing in recent years with a documented prevalence of 4 to 7 percent of the general population.Rupture is defined as “the presence of blood outside adventitia of the dilated aortic wall.” This is a rather heterogeneous event that can result in profuse retroperitoneal hemorrhage or may be more limited by an anatomic tamponade.The role of point of care ultrasound may help fill this natural diagnostic gap. Because clinicians must maintain a broad differential diagnosis in the setting of abdominal or back pain, POCUS could provide a cheap and efficient method to rule out cannot miss diagnoses. Nixon et.al have reported diagnosing AAA using POCUS with the 100% sensitivity and 93% specificity. In conclusion, this case highlights the importance of a thorough history and high suspicion in the setting of a patient with risk factors for AAA presenting with abdominal pain. Although traditionally clinicians may have been taught to expect marked hypotension or hemodynamic instability in patients with ruptured AAA, relative hypotension or normal blood pressures are fairly common at presentation. Therefore, clinicians must maintain a broad differential in this setting. Now and in the future, POCUS should be a major tool for clinicians in this challenging decision-making process to assure such a devastating diagnosis will not be missed.


The association between adult-onset Still’s disease (AOSD) and malignancy has previously been observed. However, only a limited number of cases described a combination of AOSD and leukemia, none of which reported AOSD-related symptoms as the first manifestation of acute myeloid leukemia (AML). This presentation might represent a paraneoplastic syndrome or leukemic arthritis mimicking AOSD.Here the authors report a case of a 23-year-old female who fulfilled the Yamaguchi criteria for an AOSD diagnosis. She presented with complaints of polyarthritis, sore throat, and daily fever spikes with the appearance of a nonpruritic maculopapular salmon-colored rash. Her laboratory work showed marked pancytopenia, which led to a bone marrow examination and an AML diagnosis. The patient started receiving chemotherapy with considerable improvement in the AOSD-related symptoms.Patients with underlying malignancies could present with systemic features compatible with AOSD, which necessitates excluding malignancy in any patient with this presentation, specifically in light of some warning signs like pancytopenia.This case interprets a rare association between AOSD and AML. In addition, it highlights how crucial it is to be aware of the signs that should warn the clinician of a possible underlying malignancy in any patient presenting with AOSD-related symptoms. HighlightsPatients with underlying malignancies could present with adult-onset Still’s disease (AOSD) symptoms.Clinicians should be aware of the warning signs in AOSD patients.Leukemic arthritis could mimic AOSD.AOSD-associated leukemia might represent a paraneoplastic syndrome.First coined by EG Bytwars in 1971, the term ‘adult-onset Still’s disease’ (AOSD) is used to refer to a rare systemic condition categorized as a multigenic autoinflammatory disorderThere is no specific test for the diagnosis of AOSD. However, there are several proposed classification criteria sets, of which the Yamaguchi criteria are the most commonly used ones Table . To consEven though the association between AOSD and malignancy has previously been observed, the relationship underlying this association is not entirely understoodThis case report has been reported in line with the SCARE 2020 criteriaA 23-year-old female presented to the rheumatology clinic with a 2-month complaint of symmetric polyarthritis involving the elbows, wrists, knees, and ankles, accompanied by a daily fever and significant weight loss. She reported that her symptoms were first associated with a sore throat, which had improved about 2 weeks before the presentation. She had no past morbidities or prior surgeries and did not smoke tobacco or drink alcohol. Her family history revealed that her younger brother was diagnosed with neuroblastoma at the age of four. She had received paracetamol for the past 2 months before the presentation and reported a resolution of her fever upon taking the medicine but no improvement in her pain. On examination, her heart rate was 117 beats per minute, her respiratory rate was 17 breaths per minute, and her blood pressure was 100/70 mm Hg. The patient was pale, irritable, and looked in pain. Her cardiac and pulmonary examinations were within normal limits. Her joint examination was positive for swelling and tenderness in her wrists, knees, and ankles. There was no lymphadenopathy, hepatomegaly, or splenomegaly. She was admitted to the rheumatology department for observation and further investigation. Her temperature chart showed a daily fever spike twice a day, with peaks reaching 40°C. On the second day of the admission, she developed a nonpruritic maculopapular salmon-colored rash during the febrile episodes (Fig. Laboratory data at the time of presentation are summarized in Table The association between AOSD and malignancy has previously been observed.A solid tumor was diagnosed in half of the reported cases, with breast, lung, and thyroid cancers being the most frequent typesAOSD-associated leukemia is rare and its cause remains unclear. The temporal relationship between these two entities plays a significant role in predicting the etiology underlying this association. Nakagawa and Sugawara published two cases of AOSD-associated leukemia, in which the diagnosis of AOSD preceded the diagnosis of leukemia by 2 and 6 years, respectively.They both suggested that the cyclosporine used in AOSD treatment might have induced the development of leukemiaet al.Most of the AOSD classification criteria share the requirement of excluding malignancy before the final diagnosis. However, there are no obvious guidelines regarding how strict the application of this exclusion criterion should be. Hofheinz Our approach was limited by the unavailability of both the cytokine tests and the glycosylated ferritin level test, which is a more specific marker for AOSD than serum ferritinWe report a case in which a presumptive diagnosis of AOSD was made with simultaneous detection of AML and demonstrated the significance of further investigation in any patient with AOSD-related symptoms in order to exclude any underlying malignancy, specifically in light of some warning signs like pancytopenia.None.Written informed consent was obtained from the patient for the 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.None.R.I.: literature review, manuscript writing and editing, review and approval of the final manuscript. T.D.: obtaining informed written consent, clinical follow up, manuscript writing, and approval of the final manuscript. Z.S. and H.A.: manuscript writing and approval of the final manuscript. M.K.: mentor and approval of the final manuscript.All authors declare no conflict of interest.This case report do not detail a new surgical technique or new equipment/technology, this registration was not required.Ranim Ibrahim.Not commissioned, externally peer-reviewed.


Polymorphous adenocarcinoma is a rare neoplasm of the minor salivary glands with an indolent behavior. Here, we report and describe the computed tomography (CT) and magnetic resonance imaging (MRI) of polymorphic adenocarcinoma in a 69-year-old patient who presented with local recurrence seven years after the initial treatment. On contrast CT, the primary lesion appeared heterogeneous and invaded the pterygopalatine fossa and the sphenopalatine foramen. The recurrent lesion on MRI showed a hypointense signal on the T1-weighted sequence, a hyperintense signal on the T2-weighted sequence, and heterogeneous enhancement with contrast. The patient underwent a new surgery for lesion resection and is currently under clinical and radiological follow-up. It is recommended to follow patients for at least 15 years after diagnosis, as local recurrences can occur up to 10 years after the initial treatment. Salivary gland tumors are rare, accounting for approximately 8% of head and neck tumors . PolymorA 69-year-old female patient presented with an enlargement in the right maxillary region associated with pain and reduced mouth opening. The symptoms had progressively worsened over the last five months. The patient denied smoking and alcoholism. During the physical examination, a fixed mass was palpated in the region of the right maxilla. Computed tomography (CT) of the neck showed a large solid expansive mass without calcifications inside, with heterogeneous contrast enhancement. The mass compromised the maxillary sinus, maxilla, and right nasal fossa, with diffuse bone remodeling and multiple small areas of cortical ruptures. The mass was well delimited, with numerous hypodense, irregular areas of varying sizes that insinuated into the lower thirds of the orbit, the ethmoid, and the roof of the oral cavity to the right and the left nasal cavity. The lesion infiltrated the pterygopalatine fossa and sphenopalatine foramen, destroyed the pterygoid plates on that side, and bulged the rhinopharyngeal wall in contiguity, with a blurring of the tubal torus Figure , with noThe patient underwent a total right maxillectomy, and histopathological analysis of the specimen confirmed the diagnosis of PAC originating in the minor salivary gland. The tumor had infiltrated connective tissue, bone, and perineural tissue, but no vascular invasion was observed. The patient remained under follow-up once a year for seven years until she complained of pain in the infranasal and left maxillary region. CT Figure  and magnSalivary glands are exocrine structures responsible for producing and secreting saliva and are divided into two groups, namely, major and minor. The major salivary glands are the parotid, submandibular, and sublingual glands. The minor salivary glands are acini that are distributed in the mucosa of the oral cavity and pharynx, mainly in the transition between the hard and soft palate. The control of salivary secretion of the minor salivary gland is carried out by the pterygopalatine ganglion, optic ganglion, and submandibular ganglion .Salivary gland tumors are a rare and heterogeneous group of lesions, accounting for approximately 2.6%-8% of head and neck tumors in adults ,5,6. PACPRKD gene rearrangements; and may have a target appearance on histopathology [PAC presents with cytological uniformity, morphological diversity, and an infiltrative growth pattern; stains positive for S100 on pathology; has Lesions originating from minor salivary glands require imaging examinations to confirm their presence and determine their characteristics and extension . PAC imaIn the radiological investigation of salivary gland tumors, other differential diagnoses should be considered. Among benign pathologies, pleomorphic adenoma stands out, which on MRI shows a hypointense signal on T1-weighted sequences, a hyperintense signal on T2-weighted sequences, and usually exhibits homogeneous enhancement. In the group of malignant neoplasms, the differential diagnosis should be made with adenoid cystic carcinoma. On imaging, adenoid cystic carcinoma presents as an intermediate-to-low signal on both T1 and T2-weighted sequences. Mucoepidermoid carcinoma often demonstrates a low-to-intermediate signal on both T1 and T2 imaging, with cystic components appearing more T2 hyperintense. Acinic cell carcinoma shows a hyperintense signal on T2-weighted sequences for cystic/necrotic components, and the solid component exhibits a mildly hyperintense signal on T2-weighted sequences .Surgery is the preferred treatment, with complete excision of the lesion. In the presence of bone invasion of the palate, maxillectomy is performed to a variable extent, with immediate reconstruction to maintain functionality or after a disease-free period. Lymph node dissection can be performed if alterations in lymph nodes are identified on imaging examinations . RadiothFollow-up of patients can be routinely performed with clinical examinations and MRI  for a peThis report describes the radiological and pathological features of a rare case of PAC with metastasis occurring seven years after primary tumor resection. It also highlights that the radiological diagnosis can be challenging and may be confused with other more common tumors because it presents with low signal on T1-weighted sequences, hyperintensity on T2-weighted images, and homogeneous enhancement on post-contrast T1-weighted MRI images, as well as with heterogeneous contrast enhancement on CT. Furthermore, it emphasizes that follow-up of patients with this neoplasm should be conducted with regular clinical and radiological examinations for the diagnosis of local recurrence for a minimum period of 15 years.


Background: Foot drop (FD) is characterized by an inability to lift the foot against gravity because of dorsiflexor muscle weakness. The aim of the present study is to report a clinical case of acute non-traumatic FD in patients with peroneal intraneural ganglion, after performing a scoping review on the methodological management of this disease. Methods: We performed a review of the literature and reported the case of a 49-year-old man with acute FD caused by an intraneural ganglion cyst of the peroneal nerve. Results: Out of a total of 201 articles, 3 were suitable for our review beyond our case report. The acute FD caused by peroneal intraneural ganglion can be managed by a careful clinical–instrumental differential diagnosis. A targeted surgery with subsequent rehabilitation produced a satisfactory motor recovery. Conclusions: Acute FD requires an appropriate diagnostic–therapeutic framework to identify and effectively treat the causes in order to promote complete recovery Foot drop (FD) is a clinical impairment characterized by an inability to lift the foot against gravity because of a dorsiflexor muscle weakness . PatientThe aim of the present study is to report a clinical case of acute non-traumatic foot drop in patients with peroneal intraneural ganglion, showing our management and performing a scoping review on the diagnostic therapeutic procedures of this disease.A literature review was performed, including all articles referred to patients (with an age range of 0 to 99 years) affected by acute nontraumatic FD secondary to peroneal nerve intraneural ganglion cyst. Moreover, a clinical case concerning the diagnostic therapeutic management of our patients with same disease was also presented. The review was structured in accordance with the guidelines dictated by the “PRISMA statement 2020”   query was formulated, the strings were built using the (MESH) database and Boolean indicators by associating all the possibilities of interaction between the various synonyms of the keywords, such as “peroneal ganglion”, “ganglion cyst”, “peroneal intraneural ganglion” and “foot drop”. Articles that did not fit our research question, for example, describing only non-acute-onset cases, post-traumatic cases, articles not accessible or not in English, as well as incomplete and duplicate studies, were excluded.The sequence of steps used in the review is summarized by the QUORUM flowchart . A totalA 49-year-old man, a worker, was evaluated to our outpatient clinic of Second Neurology at the University of Campania “Luigi Vanvitelli”, in November 2022. He reported a severe weakness of the ankle-dorsiflexor muscle associated to paresthesia of the lateral region of the right leg, occurring on the morning with an acute onset, about ten days ago, while getting into a car. He also reported a remote pathological history of arterial hypertension treated by beta-blocker and ACE-inhibitor drugs, a previous episode of left lumbosciatica, no further significant previous pathologies, no family history of specific pathologies and no previous or recent traumatic events. The patient underwent magnetic resonance imaging of the lumbosacral spine, which showed “severe disc-osteophytic protrusions L3/L4, L4/5 and L5/S1 with diffuse arthritic thickening of the facet joints and yellow ligaments on the arthritic-interpophyseal base, with consequent stenosis of the canal and neuroforamina”. He was previously treated by a neurological colleague with NSAIDs, Pregabalina and corticosteroids for “acute right lumbar sciatica”. After 10 days of treatment without beneficial effects, the patient came to our outpatient clinic, where a careful clinical examination revealed paresthesia in the lateral region of the right leg with associated pain symptoms  8/10), right foot drop with significant strength deficit of the tibialis anterior (TA) muscle (MRC = 1/5), of Extensor Digitorum Common (EDC) and Extensor Hallucis Longus (EHL) muscles (MRC = 0/5) and of peroneal muscles (MRC = 2/5), while the strength of all other lower limb muscles was normal (MRC = 5/5). Moreover, the active Range of Motion (RoM) of the ankle was 25° of plantiflexion, with an inability for dorsiflexion a. NegatiOpen excision was performed due to important motor and axonal deficits, acute onset of symptoms, low tendency for symptom regression, high recurrence rate with voiding and corticosteroid infiltration and no response to the rehabilitation treatment with electrostimulation and physiotherapy. Informed consent was obtained from the patient for this suggested treatment. A curved incision extending proximally and distally from the superior tibiofibular joint was, therefore, applied, sufficient for complete exposure of the peroneal nerves, which were identified and isolated e,f. AfteAt clinical examination, two months after surgery, the patient showed a significant improvement in NRS = 0/10, in MRC of TA = 5/5, EDC and EHL = 4/5, and in peroneal muscle MRC = 5/5, increase in ankle dorsilexion and metatarsophalangeal joint b and absFoot drop represents a complex pathological condition, requiring a multidisciplinary approach for appropriate evaluation and treatment . When coAccording to Consales et al. , a reducThe limitations of this case report may concern the early treatment chosen without guidelines on the acute foot drop. The authors aimed to describe the good clinical outcomes after integrated surgical–rehabilitative treatment, in order to propose a reference diagnostic–therapeutic procedure helping other clinicians. Obviously, further studies on a larger patient population are needed.The appearance of acute non-traumatic foot drop secondary to IG remains a rare, dramatic event for patients and requires an appropriate diagnostic–therapeutic framework. IG should be considered in the differential diagnosis of acute foot drop. Ultrasound, MRI, EMG and ENG examinations are valuable instrumental diagnostic elements that contribute to the diagnostic classification. Although we believe that therapeutic timing plays a decisive role in motor recovery, even long-standing cases could benefit from appropriate surgical–rehabilitative treatment.


Gastrointestinal stromal tumors (GISTs) are rare tumors accounting for 0.1-3% of gastrointestinal (GI) neoplasms. In the past, GIST was classified as leiomyomas, leiomyosarcomas, and leiomyoblastomas. However, now it is evident that GIST is a separate tumor entity, and it is the most frequent sarcoma of the GI tract. We report a case of a 57-year-old female with a five-day history of black tarry stools, two episodes of vomiting of dark-colored blood, dizziness, abdominal pain, night sweats, and palpitation, provoked by a change of position. After a computerized tomography (CT) of the abdomen and pelvis, a GIST was suspected, which was confirmed with histopathology. Acute upper GI bleeding is a rare presentation of GIST. Clear guidelines should be developed for GIST. An early diagnosis is crucial for a better prognosis. Gastrointestinal stromal tumors (GIST) are rare tumors. They are considered a subtype of the mesenchymal tumors of the gastrointestinal (GI) tract accounting for 0.1-3% of GI neoplasms. GISTs were believed to arise from smooth muscles. However, evidence indicates that these tumors originate from stem cells that differentiate into the interstitial cells of Cajal, or directly from the interstitial cells of Cajal . The mosHistoryWe report a case of a 57-year-old female who presented to a private hospital with a five-day history of acute black tarry stools, two episodes of vomiting of dark-colored blood, dizziness, abdominal pain, night sweats, and palpitation, provoked by a change of position as laying down improved these symptoms. According to the patient, the symptoms were relieved by lying down and drinking water. During the hospital admission, she also reported developing stabbing abdominal pain that was mildly relieved with water intake and associated with nausea and multiple episodes of extensive black bloody vomiting. The patient reported a history of weight loss (9kg) since the onset of symptoms. She had a past medical history of hyperlipidemia. Her past surgical history was not clear. She denied any recent travel, contact with sick patients, smoking, and the use of alcohol or drugs. The patient developed uncontrollable smelly bloody diarrhea. The patient denied any history of heartburn, pain radiating to the back, or pain aggregated or relieved by meals.ExaminationThe patient was vitally and clinically stable. The patient was afebrile, pallor was not noted on the skin or the eyes. On examination, the patient had mild epigastric abdominal pain and tenderness. The patient had no signs of jaundice, encephalopathy, edema, or caput medusa, which excluded liver disease or portal hypertension. There was no abdominal destination, or rebound tenderness. The bowel sounds were normal. The cardiac examination was insignificant with a normal S1 and S2.InvestigationsThe patient was suspected to have a GIST. An endoscopy was used to provide an accurate evaluation, which indicated an isolated fundus varix and non-active esophageal varix.A computerized tomography (CT) of the abdomen and pelvis was done, and the results showed a submucosal/endophytic gastric fundus heterogeneously hyperdense structure Figures -2. A CT The diagnosis was confirmed through magnetic resonance imaging (MRI) of the abdomen, indicating GIST Figure .We received this specimen Figure  in formaImmunobiological staining was used to differentiate between the types of GIST. In our patient, the caldesmon stain was positive, which is only observed in 10% of all the cases Figures , 6, 7, 8The neoplasm is positive for DOG1, Caldesmon, and CD117. It's negative for Ki-67.Surgical intervention, outcome, and follow-upConsidering the radiological and histopathological findings, the patient underwent a laparoscopic partial fundus resection as it was appropriate for this case. The tumor was resected to a negative margin of 1 cm distal to the mass. The patient tolerated the procedure well and was transferred to the post-anesthesia care unit (PACU) in stable condition. The patient was discharged in a stable condition and instructed to come to the ER in case of severe pain, fever, or vomiting. An outpatient appointment was scheduled after six months for clinical and radiological follow-up.GISTs, previously classified as leiomyosarcomas due to their resemblance to smooth muscles, based on immunohistochemistry, have been recognized as stromal tumors associated with the expression of neural crest cell antigens in 1984. The cellular origin of GITS is thought to be from mesenchymal stem cells, programmed for differentiation into the interstitial cell of Cajal. These cells initiate coordinated GI motility and are the pacemakers of the GI system . To currDiagnosisThe location of GIST impacts the diagnostic workup involved. In all patients, regardless of the presenting symptoms, a history and a physical examination should serve as the starting point for the diagnostic work-up. Most frequently, patients with these tumors present with anemia and other signs and symptoms of chronic GI bleeding. Patients rarely present with acute GI bleeding manifesting as melena or hematochezia . We beliTreatmentThe treatment approach for GI stromal tumors depends largely on the size and stage of the tumor. When dealing with a localized tumor that can be surgically removed and is larger than 2 cm, the primary treatment method is surgical resection . HoweverPrognosisThe outlook for individuals diagnosed with GI stromal tumors depends on factors such as where the tumor is located, the rate of cell division (mitotic count), and the size of the tumor. Additional factors that influence the prognosis include whether the tumor is completely removed with no residual cancer cells at the surgical margins and whether there was a rupture of the tumor during the surgical removal. Common complications associated with GI stromal tumors include GI bleeding and the physical pressure exerted by the tumor. Chronic GI bleeding caused by GIST is the most frequent complication and can lead to anemia. Furthermore, these tumors can also result in intestinal blockage, bleeding within the abdominal cavity, and rupture followed by inflammation of the abdominal lining (peritonitis) .Mixed GISTs are rare, especially when presenting as acute GI bleeding. GIST presenting acutely can be misdiagnosed initially as isolated gastric fundal varices. Radiological imaging plays a major role in diagnosing the condition. Different treatment options are considered, such as medical and surgical therapy. Although the mechanism behind the development of GVs is known, the association between GVs and GIST needs further studies in terms of prevalence, management, and complications.


Caruncle dysgeneses are extremely rare and must be differentiated from caruncular and conjunctival tumors. Very few case reports with histopathological descriptions exist. In this case series, four patients with five caruncle dysgeneses, two with histopathological findings, are characterised.Patient 1, a 26-year-old woman, presented with a conjunctival change at the left lower eyelid she had first noticed seven months earlier. She reported foreign body sensation and itching. On her left eye was a subtarsal conjunctival tumour measuring approximately 4 × 4 mm with whitish sebaceous gland-like inclusions located almost in the fornix morphologically resembling the nearby caruncle. The patient was asymptomatic after excision. Histopathological examination of the excised tissue showed non-keratinizing squamous epithelium with goblet cells. Subepithelially, there was lymphoplasmacytic cellular infiltration with intervening epidermal cysts adjacent to sebaceous glands and underlying adipose tissue, but no hair follicles or sweat/lacrimal glands. The epidermal cysts contained scattered hairs. A diagnosis of supernumerary caruncle was made.Patient 2, a 56-year-old woman, was referred for evaluation of a caruncle tumour that was reported to be present since childhood. Clinically the 5 × 5 mm measuring tumour appeared yellowish and less reflective compared to the normal caruncle tissue. Histopathologically, non-keratinizing squamous epithelium with goblet cells was found. In the area of more exposed tumour tissue, there were significantly fewer goblet cells and incipient keratinization of the superficial epithelial layers. Subepithelially, sebaceous glands and adipocytes were present. Hair follicles or sweat/lacrimal glands were not evident. A diagnosis of megacaruncle was made.Patient 3, a 58-year-old woman with Goldenhar syndrome, was clinically diagnosed with a supernumerary caruncle on the right eye as an incidental finding.Patient 4, a 24-year old man, clinically presented with a megacaruncle on the right eye and a supernumerary caruncle on the left.Caruncle dysgeneses are often asymptomatic and have to be differentiated from other caruncular and conjunctival tumors. If they are present, attention should be paid to signs of an oculo-auriculo-vertebral spectrum as Goldenhar syndrome. In case of unclear findings or complaints, excision with subsequent histopathological examination is required. It lies adjacent to the acute-angled fusion of the upper and lower eyelids. Laterally, the caruncle is bordered by the plica semilunaris. The caruncle consists of conjunctiva, hair follicles, sebaceous glands, and accessory lacrimal glands. Caruncle tumors, neoplastic and non-neoplastic, have already been described.A keyword search was performed in the database at the Department of Ophthalmology, University Hospital Freiburg, using the word “caruncle” from January 1, 2006, to May 19, 2022. Where available, all written and photographic documents were examined for evidence of caruncle dysgeneses. The localization, subjective complaints, histological characteristics, and measures performed were taken from the records. Additionally, we conducted a literature research via MEDLINE with the search terms “supernumerary caruncle”, “ectopic caruncle”, “megacaruncle”, “lacrimal caruncle dysgenesis” and “caruncle malformation”. Written informed consent for publication of their clinical details and clinical images was obtained from the patients. Approval was given by the local Ethics Committee of the University of Freiburg under the following number (22/15).22.12.1.1A 26-year-old female patient was referred to the Eye Center for co-evaluation of a conjunctival tumor and suspected pyogenic granuloma on the tarsal lower lid of the left eye (LE). The patient reported that the conjunctival change was first noticed seven months earlier and was initially treated with lubricating eye drops. Subjectively, the findings had been stable in size during the last seven months. There was no bleeding. The patient suffered from foreign body sensation and itching at the LE. Currently, there was no ophthalmologic therapy.2.1.2Best corrected visual acuity (BCVA) on both eyes (BE) was 20/20 (right eye (RE) +0,5/−1,0/178°, LE +0,25/−0,25/178°). Intraocular pressure (IOP) was normotensive at 16 mm Hg bilaterally. The RE showed no abnormalities of the conjunctiva or the anterior segment except for subtarsal follicles. On the LE, there was a subtarsal conjunctival tumor measuring approximately 4 × 4 mm with whitish sebaceous gland-like inclusions located almost in the fornix, which morphologically resembled the nearby caruncle A. HoweveWith subjective complaints at the LE and atypical findings for a pyogenic granuloma, we decided together with the patient to excise the conjunctival tumor. Subsequently, a therapy with ofloxacin eye ointment three times daily for five days was recommended. Four weeks postoperatively, the patient reported a decrease of the previously described symptoms.2.1.3Histopathological examination of the excised tissue was performed by haematoxylin-eosin (HE) staining B&C and p2.22.2.1A 56-year-old female patient was referred for evaluation of a caruncle tumor on the nasal corner of the lid of the LE. The patient reported that the change had been present since childhood and had grown over the past one to two years. Subjectively, the patient did not feel affected by the tumor on her LE.2.2.2BCVA on the RE was 20/20  and on the LE 20/25 . IOP was normotensive on BE (18/19 mm Hg). The RE showed no abnormalities of the conjunctiva, caruncle, or anterior segment except for a prolapse of fatty tissue. The LE showed a caruncle tumor adjacent to the normal caruncle that appeared to be fused to the medial lid ligament A&B. On s2.2.3In the histopathological examination, HE staining and PAS reaction revealed non-keratinizing squamous epithelium that was four to six cells thick and had an admixture of goblet cells C–E. In t2.3A 58-year-old female patient with known Goldenhar syndrome presented. BCVA was 20/30 (+1.5/−0.75/46°) on the RE and 20/20 (+0.5/−0.75/14°) on the LE. She was diagnosed with a supernumerary caruncle on the RE as an incidental finding . In addi2.4A 24-year-old patient presented because he had noticed a change on both lower eyelids  without 2.5Four cases of caruncle dysgeneses are presented. At the time of diagnosis of caruncle dysgeneses, all four patients were adults. The ratio of female to male patients was 3:1. Two cases involved the LE, one the RE, and one both eyes. One patient with a supernumerary caruncle reported foreign body sensation and itching. All other patients were asymptomatic. Three times a diagnosis of supernumerary caruncle and two times a diagnosis of megacaruncle was made. One patient with a supernumerary caruncle had an association with Goldenhar syndrome .Table 1C3In the present case series, four cases of the rare caruncle dysgeneses are described.1.supernumerary caruncle is a rare congenital anomaly. In addition to the normal caruncle, additional tissue unrelated to the normal caruncle is present, showing histologic features similar to those of the latter. It may occur on the lower or upper medial third of the eyelid and may be single or multiple.The The exact embryological development of the caruncle has not been conclusively clarified. From the shiny surface of the orthotopic caruncle, 15 to 20 small lobules of sebaceous glands shimmer through the conjunctiva and fine, almost transparent lanugo hairs grow from hair follicles. Histopathologically, the caruncle-plica complex is covered by a goblet cell-rich, nonkeratinizing squamous epithelium. In addition to the hair follicle-gland units, PAS-positive lobules of accessory lacrimal glands may be evident. In a review by Jakobiec et al. several dysgeneses of the caruncle are described, which have to be distinguished from each other,,2.ectopic caruncle. In contrast to the aforementioned supernumerary caruncle, a true ectopic caruncle is usually dysplastic and refers to a displacement of a tissue remnant from its normal commissural anatomic location, always at the medial posterior aspect of the lower eyelid. Such an anomaly may be associated with a number of other diseases of the ocular appendages. No case of ectopic caruncle was found in our case series.The 3.Megacaruncle describes a considerable enlargement of the caruncle. In contrast to the supernumerary caruncle, there is a continuous connection of the additional caruncle tissue with the orthotopically located caruncle.Given the normal caruncles on both eyes and the distinction of the caruncle from the tumor tissue by normal conjunctival tissue, our first case is most likely a supernumerary caruncle, even though no hair follicles and lacrimal glands were histologically visible. Both the macroscopic aspect and the histological finding of sebaceous glands not typically found in the conjunctiva of the fornix, support this diagnosis. In addition, hair was found in the epidermal cysts and lead us to the assumption that these epidermal cysts could have grown in association with rather abnormal hair follicle development. Although the patient did not notice the supernumerary caruncle until the third decade of life, it can be assumed that it had existed since birth and was only discovered by the new onset of symptoms with foreign body sensation and itching. In the three cases of a supernumerary caruncle described in the literature, it was documented that the eye irritation was caused by surface irregularities and lanugo hairs; the symptoms disappeared after surgical excision.In our second case with the diagnosis of a megacaruncle, a choristoma , but also true neoplasms of the caruncle can be considered as differential diagnosis. In a retrospective evaluation of 82 caruncle tumors at the University Eye Hospital Bonn between 1998 and 2020, one case of a caruncle choristoma, a dermoid, was described.The oculo-auriculo-vertebral spectrum (OAVS) is a congenital disorder of craniofacial morphogenesis. It is characterized by malformations of structures derived from the first and second branchial arches, including the eyes, lips, tongue, palate, ear, maxilla and mandible. Goldenhar syndrome is part of the OAVS and describes mostly more severe manifestations.Due to the association of malformations of the caruncle with Goldenhar syndrome, it is reasonable to assume that a disorder in the formation of the first and second branchial arches is responsible for the malformation even in otherwise healthy patients. Although the malformation of the caruncle is present from birth, it is usually not diagnosed until adulthood. Apart from cases in children with OAVS, only one case of a supernumerary caruncle in a 12-year-old child has been published so far.4In case of subjective complaints, size progression, changes or suspicion of malignancy, excision of the tissue with histopathological examination should always be performed. In most cases, the procedure can be performed under local anesthesia. Long-term surgical sequelae are not expected. Symptoms such as itching, foreign body and pressure sensation can be alleviated if the tumor is their cause. If the patient is symptom-free, the findings are stable in size, the slit-lamp microscopy is clear and there are no indications of malignancy, a “watch and wait” approach with regular ophthalmologic examinations may be considered.Written informed consent for publication of their clinical details and clinical images was obtained from the patients.CJW and CAH contributed to the study conception and design. Material preparation, data collection and analysis were performed by CJW. The first draft of the manuscript was written by CJW and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.10.13039/501100002714University of Freiburg.We acknowledge support by the Open Access Publication Fund of the CJW had full access to all the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.The data presented in this study are available from the corresponding author upon reasonable request. The data are not publicly available due to privacy and ethical issues.Approval was given by the local Ethics Committee of the University of Freiburg under the following number (22/15).Written informed consent for publication of their clinical details and clinical images was obtained from the patients.The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.


Giant cell tumor of the bone (GCTB) is a benign bone tumor that can occasionally progress to malignancy, usually in chronic cases. It is a common benign and aggressive bone tumor that affects patients aged between 20 and 45 years. The most common location is the knee joint. It manifests as a painless or occasionally painful swelling over the affected area. A case of giant cell tumor (GCT) of the proximal tibia in a 72-year-old male is reported here, which was difficult to diagnose as it is rarely found in the geriatric age group. The patient came with a chief complaint of pain and swelling over his left knee for two months with a history of trauma to the knee a couple of times. On clinical examination, the patient had grade 3 tenderness and swelling on the anterolateral aspect of the knee extending toward the proximal tibia. The swelling was well-defined, smooth, firm, and uniform in consistency with dimensions of 15 cm × 12 cm. The swelling was moveable sideways, and the movement of the knee suggested that it was not attached to the underlying bone. As per the age and history of the rapid-growing lesion, we suspected malignancy, but clinical findings were pointing toward benign tumor. X-ray of the affected knee was done, which revealed a soft tissue mass with the involvement of the bone. Magnetic resonance imaging (MRI) of the knee revealed a soft tissue mass with the cortical breach. An open biopsy was done for the confirmation of the diagnosis, which was later reported and confirmed as a giant cell tumor of the proximal tibia. As bone tumor is associated with a cortical breach and pathological fracture, it was classified under Campanacci grade 3, for which an en bloc resection and open reduction and internal fixation with plate osteosynthesis with bone cementing and bone grafting were done followed by knee bending physiotherapy and gradual weight-bearing. Finally, the knee function was improved with pain relief. Although giant cell bone tumors are typically benign, they can occasionally be malignant and locally aggressive ,2. Bone A 72-year-old male obese patient with a history of systemic hypertension, who is a farmer by occupation, visited the orthopedics department with chief complaints of pain and swelling over the left knee for two months with a history of falls for two times in the last 20 days and an inability to bear weight since the last 20 days. The pain was associated with swelling, which was insidious in onset and gradually progressive in nature. The pain and swelling occurred without any prior trauma. The swelling grew in size over time, and the pain got worse when standing or walking. The pain aggravated after the fall, and the patient was unable to bear weight. The pain interferes with the patient's daily activities. The patient was advised to do some investigations such as X-rays Figure , MRI , and through the semisolid cement, a lateral locking proximal tibia plate was inserted. The excised tumor was sent for histopathological examination, which showed polygonal to round histiocytes surrounded by multinucleated giant cells, fibro-fatty tissue Figure  suggestiPostoperatively, the physiotherapy treatment advised to the patient was paraffin wax bath for 20-30 minutes in three sessions to help relax the muscles and reduce pain and edema. To increase the range, some range of motion exercises were given, which include active assisted hip range of motion (ROM) exercises, active ankle ROM exercises, and passive knee ROM exercises. ROM exercises were given at intervals of 10 repetitions twice a day to prevent contractures and deformity.2O2), cryosurgery, and argon beam are used to reduce recurrence. Systemic therapies such as bisphosphonates, interferon alpha (IFN-a), and denosumab can be utilized to lower the likelihood of local recurrence. En bloc resection and reconstruction are the major treatments for grade 3. Other solutions for the reconstruction exist as well, such as cannulated cancellous (CC) screws and Steinmann pins; however, plating offers higher stability and rigidity.The case reported in this article was presented to the hospital as a high-grade giant cell tumor of the left knee, which was diagnosed and confirmed on histopathological examination after the open biopsy. Excision of the tumor was surgically done with bone grafting and plating. At six weeks of follow-up, the patient does not show any clinical or radiological features of recurrence. The patient was asked to come for follow-up six weekly at least for a period of one year. Curettage was the mainstay of therapy for giant cell tumors, especially for grades 1 and 2, but associated with a high recurrence rate (35%-40%). Thus, adjuvants such as bone cement, phenol, hydrogen peroxide (HIn their study published in 2016 on Steinmann pin augmentation versus locking plate constructs, Ruskin et al. concluded that locking plate constructs had greater stiffness than tibial constructs fixed with Steinmann pins . In theiPostoperatively, follow-up was held every six weeks. The patient had complete relief of pain, with improvement in the range of movement, and knee flexion to 70 degrees with full extension without any surgical site complications evident. The patient was able to perform her activities of daily living. No evidence of recurrence was noted on clinical and radiological examination.Giant cell tumor of the bone in a left proximal tibia with a pathological fracture in an elder male was unusual. Thus, with the help of history and clinical examination with radiological and histopathological examination, we can reach a proper confirmatory diagnosis, which was helpful in proper preoperative planning, operative decision with complications, and possible outcomes.


Mirizzi syndrome (MS) is an uncommon condition caused by chronic gallbladder stones, leading to external compression and obstruction of the common hepatic duct. This report details an unusual MS case in a 65-year-old man who experienced right upper abdominal pain, jaundice, fever, and nausea. Diagnostic tests, such as ultrasound and CT scan, indicated acute acalculous cholecystitis resembling MS. However, a magnetic resonance cholangiopancreatography (MRCP) confirmed no gallstones in the biliary system. The patient's laparoscopic cholecystectomy was successful, with tissue analysis revealing intense gallbladder inflammation and epithelial necrosis but no gallstones. This case emphasizes the diagnostic complexities of atypical MS presentations and the need for comprehensive diagnostic methods, including MRCP. Additionally, the report advocates for standardized terminology in medical literature to ensure clear communication among medical professionals. Mirizzi syndrome (MS), an uncommon consequence of prolonged gallbladder stones, was first identified in 1948 . In the In the subhepatic region, severe inflammation and adhesions often affect the hepatoduodenal ligament, disrupting the typical anatomical relationships and dimensions. Acute acalculous cholecystitis (AAC) can lead to a syndrome that closely resembles multiple sclerosis in clinical progression and imaging characteristics . This meA 65-year-old man presented to the outpatient department with a 10-day history of right upper abdominal pain, which was followed by jaundice, fever, and nausea over the last four days. He had no history of diabetes, hypertension, or viral hepatitis. Initially healthy, he suddenly experienced intense, dull pain in the right upper abdomen that radiated to the mid-back, alleviated by painkillers. He later developed a fever and noticed his eyes turning yellow. He also mentioned experiencing constipation for the past three days and having no prior health concerns or notable family medical history.Diagnostic tests revealed markedly elevated total, conjugated, and unconjugated bilirubin levels, indicative of obstructive jaundice. Additionally, there were elevated white blood cell counts and neutrophilia, suggesting an underlying inflammatory process. The marked elevation in alkaline phosphatase (ALP) further pointed toward obstructive jaundice. The complete blood workup that was carried out initially is detailed in Table Following the initial findings, a more detailed assessment was conducted. An ultrasound revealed a rough liver texture and an enlarged common bile duct (CBD) without detectable stones. A positive Murphy's sign was noted, along with dilated intra- and extrahepatic biliary channels and an inconsistent liver texture. Acute acalculous cholecystitis was the initial diagnosis. However, certain clinical symptoms and ultrasound results hinted at an obstructive issue, leading to a CT scan for further clarity.The CT scan showed mild CBD enlargement with a sudden constriction near the supraduodenal area, causing dilation in the intrahepatic biliary channels. No significant mass or radiodense stone was detected in the CBD, and there was no sign of any ampullary or periampullary mass. These CT results confirmed the obstructive nature of the pathology. An ERCP was then conducted to solidify the diagnosis and determine the best course of action.During ERCP, proximal duct dilation and strictures were identified, resulting in distal CBD obstruction, without apparent mass. Notably, the CBD exhibited wall damage. These ERCP findings guided subsequent surgical planning. Magnetic resonance cholangiopancreatography (MRCP) revealed gallbladder enlargement with wall thickening, causing extrinsic compression of the CBD, with no discernible stones within the biliary system Figure .A laparoscopic cholecystectomy was scheduled to alleviate the patient's symptoms. Intraoperatively, a significantly enlarged and edematous gallbladder was observed. Histopathological examination of the excised gallbladder showed marked signs of severe acute inflammation and epithelial necrosis, notably without the presence of gallstones in the gallbladder or cystic duct. Additionally, no evidence of carcinoma was detected.After the surgery, the patient was advised on post-operative care, which included limited physical activity and a low-fat diet. The surgery's success was evident from the decreased ALP, AST, and ALT levels within a week. There were no post-operative complications. The patient is now closely monitored with regular check-ups to track long-term results and prognosis.A common hepatic duct obstruction brought on by the extrinsic compression of an impacted stone in the cystic duct or Hartmann's pouch defines the unusual illness known as MS. It appears in about 0.35% of surgical cholecystectomies . There mAn enlarged and inflamed gallbladder can externally compress the common hepatic or bile duct, leading to jaundice and exacerbating AAC. This mechanism is similar to MS when a stone blocks the gallbladder neck or cystic duct, causing external bile duct compression. Ippolito first reported this syndrome in 1993 using ultrasound (US) and endoscopic retrograde cholangiopancreatography (ERCP) . The stuIt is noteworthy to note that there is disagreement on the nomenclature used to define this illness. These three published case reports each had a different terminology employed by the writers. The ailment was referred to as "acute acalculous cholecystitis associated with common hepatic duct obstruction: a variant of Mirizzi's syndrome" in Ippolito's paper . While AIn acalculous cholecystitis cases, the diagnosis of MS is often made using percutaneous transhepatic cholangiography, US, or ERCP -10. To tIn patients presenting with symptoms suggestive of MS, particularly those manifesting jaundice, right upper abdominal pain, and imaging findings indicating biliary obstruction, a comprehensive differential diagnosis should be meticulously considered. Conditions such as cholangiocarcinoma, choledochal cysts, primary sclerosing cholangitis, and impacted stones in the common bile duct are prominent differentials. Each of these conditions can manifest with overlapping clinical and radiological features, further complicating the diagnostic evaluation. Notably, gallbladder pancreatic heterotopia stands out as a rare differential. This involves the ectopic location of pancreatic tissue within the gallbladder, independent of anatomical and vascular links to the primary pancreas. Although typically asymptomatic, any inflammation or growth within this ectopic tissue can simulate cholecystitis or gallbladder tumors . A thoroMRCP in our case supported the diagnosis of MS in acalculous cholecystitis. Therefore, we believe that MRCP should be an effective technique to detect any type of MS, including this uncommon illness of acute acalculous cholecystitis determining MS, even though more research is required to draw a firm conclusion.MS is a rare gallbladder stone complication that poses diagnostic and management challenges. This report describes an uncommon MS case where acute acalculous cholecystitis mimicked clinical and radiological signs of MS. The MRCP-confirmed absence of gallstones in the biliary system added to the diagnostic complexity. The patient's successful laparoscopic cholecystectomy highlights the value of a thorough diagnostic approach, including MRCP, to distinguish MS-induced common hepatic duct obstruction from other causes. Clinicians should recognize this atypical MS presentation for prompt, appropriate intervention. The literature's varied terminologies for this condition indicate a need for standardized naming to enhance clarity and communication among medical professionals.


Acute lymphoblastic leukemia (ALL) is a hematological cancer that can cause ocular tissue involvement. Asparaginase is a chemotherapy regimen that is commonly used in leukemia which could lead to similar ocular manifestations. We report a patient with a history of ALL for seven months on asparaginase therapy and persistent cerebral sinus venous thrombosis (CSVT) with acute venous infarction in the left frontal lobe presented with worsening vision. On examination, he had a visual acuity (VA) of (6/21) in the right eye and (6/60) in the left eye, with a mild left eye abduction limitation. Fundal examination showed bilateral prominent multilayered retinal hemorrhages and papilledema with absence of leukemic infiltration. His chemotherapy regimen was held and a one month follow up was scheduled. Follow up after one month of chemotherapy cessation showed resolution of both VA and fundal exam findings. It is crucial to differentiate between asparaginase toxicity and infiltration of the disease in ALL patients. As this would determine whether the treatment should be continued or suspended. Acute lymphoblastic leukemia (ALL) is a cancerous, hematologic, systemic disease of two types: B and T-cell . ALL claOcular tissue involvement could be observed in leukemic patients. Direct infiltration, hemorrhage, and ischemic changes are three common processes of ocular involvement . The acuWe report this case of a 15-year-old boy known case of ALL for seven months and left frontal lobe stroke for one week who presented with a complaint of bilateral gradual vision loss associated with double vision consistent with the presence of bilateral papilledema.A 15-year-old boy with a history of acute lymphoblastic leukemia for seven months and persistent cerebral sinus venous thrombosis with acute venous infarction in the left frontal lobe was referred to an ophthalmology department for evaluation of gradual worsening vision for one week. The patient's father reported no history of any eye surgeries, lasers, or injections. He has no history of eye traumas. There is a positive family history of diabetes mellitus, hypertension, pancreatic cancer, and hyperthyroidism. There is no family history of hematological diseases.The patient had decreased visual acuity of 6/21) in the right eye and (6/60) in the left eye. His intraocular pressure was 15 mmHg in the right eye and 17 mmHg in the left eye. His pupil examination was normal. His extraocular muscle examination showed mild left eye abduction limitation. The fundus examination revealed the presence of bilateral disc swelling with scattered small multilayered retinal hemorrhages in both eyes  was within normal limits. Fundal examination showed bilateral prominent multilayered retinal hemorrhages and papilledema with absence of leukemic infiltration. The patient’s asparaginase therapy was held and a one month appointment at the ophthalmology clinic was scheduled. During his follow up after one month of holding his antileukemic agent, the best corrected visual acuity (BCVA) in the right eye was (6/6), and in the left eye was 6/12). Furthermore, fundal exam showed bilateral resolution of multilayered retinal hemorrhages, as well as resolution of the papilledema (Figure . FurtherRetinal hemorrhage is a major complication that manifests in leukemic patients . It mighDubashi and Jain reported a similar case in which a 16-year-old male patient with ALL who developed cortical venous thrombosis manifesting as headaches, vomiting and multiple episodes of seizures three weeks after his exposure to asparaginase. The treatment was stopped as well, and a repeated CT brain was performed after one month of stopping the treatment showing normal results . SimilarThe importance of collaboration between ophthalmology-ocular oncology and oncology departments is crucial in differentiating between the leukemic infiltration and treatment side effects. In case of leukemic infiltration, treatment regimen should be continued though it is suspended in the incidence of treatment side effects. Retinal hemorrhage needs to be related to one of those etiologies and treated accordingly . FinallyIn conclusion, this case highlights the importance in differentiating asparaginase toxicity from infiltration of the disease in ALL patients; thus history, sequence of medications, and good comprehension of the side effects is important. As this would determine whether the treatment should be continued or suspended. Cooperation between specialties and fast access is important to avoid diagnosis and management delay.


Gallbladder paragangliomas are extremely rare with only a handful of cases reported so far. There are no definitive guidelines for the management of gallbladder paragangliomas due to their rarity. We present a case of a 53-year-old male who was found to have gallbladder paraganglioma post-laparoscopic cholecystectomy, performed for right upper abdominal pain. On review of the literature, all previously reported cases had been nonsecretory and benign. For patients who have no symptoms of secretory paragangliomas and no family history of endocrine syndromes, cholecystectomy and clinical follow-up may be a sufficient initial management following an incidental finding of gallbladder paraganglioma. Cholecystectomy is a commonly performed operation, especially in the Western population, for indications relating to cholelithiasis, such as biliary colic and acute cholecystitis . About 1A 53-year-old male presented with an eight-day history of right upper quadrant (RUQ) abdominal pain, which started suddenly and persisted. His vital signs were all within normal ranges . Abdominal examination was unremarkable with negative Murphy’s sign. Laboratory tests including complete blood examination, C-reactive protein, and lipase were all normal, deviating away from differentials such as acute cholecystitis and gallstone pancreatitis. Liver function tests were mildly deranged, with alkaline phosphatase of 183 U/L, gamma-glutamyl transferase of 237 U/L, alanine aminotransferase of 148 U/L, and aspartate aminotransferase of 115 U/L. Abdominal ultrasonography (US) showed mild hyperemia along the gallbladder wall with mobile sludge, but no other features of acute cholecystitis such as pericholecystic fluid.He was admitted for pain control and monitoring, and due to persistent RUQ pain, he was taken for laparoscopic cholecystectomy two days after presentation. Intraoperatively, the gallbladder appeared normal with only mild wall edema. His blood pressure was stable between 110 and 130 mmHg (systolic) throughout the operation and admission. He was discharged with complete resolution of pain and no perioperative complications.Macroscopically, the gallbladder contained no gallstones and had yellow mucosal flecks, which raised suspicion for cholesterolosis. The wall had a normal thickness of 2 mm. Microscopic examination revealed a single, 0.5-mm tumor centered in the adventitial adipose tissue, which consisted of a well-circumscribed aggregate of tightly packed epithelioid cells Figure . The lesParagangliomas are neoplasms of the paraganglia, originating from the neural crest cells ,3. ParagAn online search using the keywords “gallbladder,” “paraganglia,” and “paraganglioma” on the National Library of Medicine found 24 cases of gallbladder paragangliomas since 1972 (Table SDHD) gene mutation, suggested Gallium-DOTATATE positron emission tomography (PET) as an essential follow-up investigation for any patients with hereditary paragangliomas, as the gallbladder paraganglioma in this male lacked F-fluorodeoxyglucose (F-FDG) avidity but was intensely avid with Gallium-DOTATATE [Radiologically, paragangliomas do not exhibit any specific features on CT or magnetic resonance imaging (MRI). Extra-adrenal paragangliomas may show a similar appearance to a hemangioma on a CT due to its hypervascularity, and on T2-weighted MRI, it may exhibit a “salt and pepper” pattern over the regions with high and low signal intensities ,21. The DOTATATE .There are no guidelines for the management and surveillance of incidental gallbladder paragangliomas due to their rarity. On review of the literature, there are no reports of complicated postoperative recovery. The authors of a case report of hemorrhagic gallbladder paraganglioma repeated abdominal CT at six weeks postoperatively, which did not show any evidence of tumor recurrence or any significant findings; however, they did not comment on whether the CT was performed to ensure that there was no further hemorrhage or for surveillance for paraganglioma . Blood aOur patient was informed of the diagnosis, and the case was discussed in a multidisciplinary meeting. As he did not have any other symptoms and no family history of endocrine syndromes, the decision for no further investigation was made.The majority of gallbladder paragangliomas were diagnosed incidentally after cholecystectomy for abdominal pain. They were benign, nonsecretory tumors occurring in patients with no known familial multiple endocrine syndromes. Blood and urine catecholamine levels are not likely to be of any diagnostic utility, and there are no specific abdominal US, CT, or MRI features to detect metastatic, recurrent, or residual tumors. There have been a few cases of gallbladder paragangliomas occurring with other extra-adrenal paragangliomas, and these were avid on Gallium-DOTATATE PET. For patients who have no symptoms of secretory paragangliomas and a history of familial endocrine syndromes, immediate investigation with further imaging may not be required, and clinical follow-up may be adequate. Patients who have or develop symptoms suggestive of other extra-adrenal paragangliomas or have a family history of endocrine syndromes may benefit from Gallium-DOTATATE PET.


Surgery is the initial form of treatment for glioblastoma, and a maximum resection without impairing neurological function improves survival. Wounded glioma syndrome (WGS) is a clinical picture observed after the resection of high-grade tumors. This syndrome, developing within hours to a few days after glioma surgery, is characterized by hemorrhage into the postoperative cavity and cerebral edema and at times occurs in areas distant from the site of the resection, i.e., distant wounded glioma syndrome (DWGS). We report a case of a 70-year-old male presenting with acute-onset left leg weakness, with a large peripherally enhancing mass with central non-enhancement suggestive of necrosis in the right frontal lobe. A gross total resection of the tumor was done, and the histopathologic evaluation verified the diagnosis of glioblastoma World Health Organization (WHO) grade IV. During the postoperative period, he was drowsy and was able to move his right extremities. He had a series of generalized tonic-clonic seizures three hours after the operation. After eight hours, the patient became comatose with signs of increasing intracranial pressure. A cranial computed tomography (CT) scan revealed diffuse cerebral edema and hemorrhage into the operative site in the right frontal lobe, as well as subarachnoid hemorrhages in the bilateral frontoparietal sulci. There were also small hemorrhages seen in the left caudate head, midbrain, and left hemipons. Death occurred the following day. This case report demonstrates an unusual case of a WGS with a concurrent DWGS in the brainstem after a gross total resection of a frontal lobe glioma. This case shows a significantly uncommon sequela that a patient undergoing glioma surgery can present, leading to rapid deterioration and death. Resection of a glioma carries a significant risk, and its impact in the immediate postoperative period merits evaluation when planning perioperative management, taking prompt action if these syndromes occur. Glioblastomas  grade IV) are the most prevalent type of malignant primary brain tumor, accounting for the highest mortality rate among patients with primary brain tumors. They are particularly aggressive and treatment-refractory, with unfavorable prognosis  . AlthougThis case report was previously presented as a poster at the 2023 Society for Neuro-Oncology (SNO)/American Society of Clinical Oncology (ASCO) Central Nervous System (CNS) Cancer Conference on August 10-12, 2023.Clinical course and radiological examinationsThe patient was a 70-year-old Filipino male with an initial Karnofsky Performance Status (KPS) score of 100. He was independent in all activities, with no prior noticeable weakness, no behavioral or mood changes, memory lapses, or disorientation, and no sleep disturbances. He had no other known comorbidities aside from controlled hypertension, for which he was not on any medication. He primarily presented with acute-onset weakness of the left lower extremity, which was noted to progress over one day with no episodes of resolution. Over three weeks, the left lower extremity weakness was persistent with no associated numbness, headache, nausea or vomiting, blurring of vision or diplopia, dysarthria, facial asymmetry, changes in sensorium or behavior, seizures, or involuntary movements. His KPS score decreased to 70 before the consultation. The neurological examination showed the weakness of only the left lower extremity and revealed the presence of several frontal release signs, including glabellar tap, snout, palmomental, grasp, and left Babinski reflexes. He had normal sensory, cranial nerve, brainstem, and cerebellar functions.Magnetic resonance imaging (MRI) Figure  showed aThe patient underwent a right frontal craniotomy for gross total resection of the tumor Figure . The admFollowing the surgical procedure, he experienced a state of lethargy and exhibited right-sided hemiparesis. He had a series of generalized tonic-clonic seizures three hours after the operation, initially resolved with the administration of intravenous diazepam. He was then started on a continuous midazolam drip for seizure control. A computed tomography (CT) scan Figure -3D reveaHistopathologyIntraoperatively, the right frontal lobe tumor appeared as a soft gray-tan intra-axial mass with irregular borders. Microsections Figure  show a hWe report a case of an elderly male who underwent a gross total resection of glioblastoma of the right frontal lobe. During the postoperative period, he experienced seizures and signs of increased intracranial pressure, leading to a coma. Following the cranial surgery, the subsequent CT scans revealed the presence of extensive cerebral edema and hemorrhages. These hemorrhages were found not only at the surgical site located in the right frontal lobe but also in several remote regions. The postoperative complications described are consistent with the findings of WGS and DWGS, as discussed in the limited available literature on these clinical entities -9. The pWGS is recognized as a postoperative complication that may arise after a tumor has been completely or only partially removed. It involves bleeding into the tumor bed, which may occur soon after surgery despite best efforts to obtain hemostasis throughout the operation -7. The cPathophysiologyGlioblastoma is characterized by its ability to create extensive networks of microvasculature through overactivated angiogenesis . In addiComplete resection of GBM poses a significant challenge due to the tendency of glioma cells to migrate from the primary tumor and potentially infiltrate the contralateral hemisphere . There aThe DWGS seen in the patient is most likely attributable to tumor-induced coagulopathy. This suggests that changes in coagulation caused by either the tumor or the surgery contribute to the formation of multiple postoperative intracranial hemorrhages. These hemorrhages are observed bilaterally, affecting not only the site of the tumor resection but also other areas of normal brain tissue . During The primary objective of glioblastoma surgery is to achieve maximal safe resection of the enhancing solid tumor while preserving neurological function ,2,4. TheFrom a clinical perspective, it would be prudent to consider both WGS and DWGS as potential surgical complications when planning the best management approach for gliomas. It is important to discuss the possibility of these complications with patients before deciding on surgery. To mitigate neurological complications, surgical intervention should aim for complete removal of lesions situated in areas where excision would not cause further neurological impairment. Nevertheless, complete tumor resection may not be feasible with distant lesions in eloquent brain regions. In such instances where reducing novel neurological deficits is critical while simultaneously achieving the maximum extent of resection, the preferred approach is awake craniotomy with intraoperative electrical stimulation mapping . It is cIn the present case, the gross total resection of a large GBM tumor has led to a fatal combination of WGS and DWGS. Hemorrhage occurring into and remote from the surgical resection site is a rare yet potentially serious complication that warrants attention when postoperative deterioration occurs. This is especially critical when the presenting symptoms are not consistent with the area that underwent surgery. Despite extensive research into the pathobiology of GBM and the remarkable progress that has been made in its management, treating this typically fatal cancer remains a challenge. A thorough assessment of coagulation and TEG studies done perioperatively can be instrumental in preventing these potential complications. Surgeons and patients must consider WGS and DWGS as possible complications before deciding to perform a particular surgical procedure. We suggest that a discussion of these possible complications, occurring independently or concurrently, be included as part of the informed consent for the surgical procedure. These syndromes and their implications and repercussions in the immediate postoperative period must be reviewed to evaluate the risks and benefits of surgery and avoid fatal outcomes.


Diagnosing intralymphatic histiocytosis can be challenging due to its rarity. We present a case of intralymphatic histiocytosis in the upper eyelid of a Korean patient. We treated the condition by surgical debulking and intralesional triamcinolone injection.A 59-year-old man was referred to our clinic with a 7-year history of unilateral swelling in the right upper eyelid. He had previously been treated with long-term oral steroids and immunosuppressants, but his eyelid swelling persisted. Unilaterally non-pitting erythematous edema was localized on the right upper eyelid without any itching or pain. His best corrected visual acuity at presentation was 20/20 for both eyes. Enhanced orbital computerized tomography revealed edematous soft tissue thickening in the right upper eyelid. In the laboratory testing, the erythrocyte sedimentation rate showed an increase of 19, and the antinuclear antibody titer was positive with a homogeneous pattern.We diagnosed the patient with intralymphatic histiocytosis based on the histopathological findings.We attempted surgical debulking and biopsy on the right upper eyelid due to the persistent symptoms and the absence of a definitive diagnosis.The patient has demonstrated significant improvement after receiving an intralesional triamcinolone injection in the right upper eyelid following the surgery and is currently under follow-up with no signs of recurrence.Ophthalmologists should consider intralymphatic histiocytosis in cases of persistent eyelid swelling that do not respond to treatment, even in Asian patients. Surgical debulking and intralesional triamcinolone injections may be beneficial for improvement. The pathogenesis of intralymphatic histiocytosis appears to be associated with chronic inflammation of the affected area, although the exact cause is undetermined. Intralymphatic histiocytosis results in lymph stasis and the subsequent development of lymphangiectasis. It has sometimes been discovered as an incidental histopathological finding. Here, we report a case of intralymphatic histiocytosis presenting as isolated unilateral eyelid edema in a patient of Korean descent.Intralymphatic histiocytosis is a rare skin condition characterized histopathologically by prominently dilated lymphatic vessels. It can clinically present with nonspecific cutaneous symptoms. It is difficult to diagnose a condition in which eyelid edema is the only clinical manifestation because it is very rare for periocular swelling to be the sole symptom.A 59-year-old man was referred to our clinic with a 7-year history of unilateral swelling in his right upper eyelid. The patient, of Korean descent, had a history of well-controlled hypertension. He had previously received oral steroid and immunosuppressant treatments at other clinics , but the swelling of his eyelids did not improve. Physical examination revealed unilateral erythematous swelling on the right upper eyelid without any itching or pain Fig. . On the Due to the persistent edema without a definitive diagnosis, an oculoplastic surgeon attempted surgical debulking and biopsy of the right upper eyelid. Hematoxylin and eosin staining of the specimen revealed a normal epidermis and papillary dermis but prominently dilated vascular structures in the reticular dermis. The dilated lymphatic lumen was filled with a chronic granulomatous inflammatory infiltrate of mononuclear epithelioid cells Fig. A and B. The patient was diagnosed with intralymphatic histiocytosis, and the symptoms partially improved after surgery Fig. A. The pa is an uncommon reactive condition clinically characterized by nonspecific cutaneous manifestations and histopathologically characterized by mononuclear histiocytes accumulation within dilated lymphatic vessels. Numerous cases of intralymphatic histiocytosis have been reported in association with various inflammatory and neoplastic diseases, including rheumatoid arthritis, reactions to metal joint implants, Merkel cell carcinoma, breast cancer, and colon cancer.,5 Allergies, chronic inflammation, and lymphangiectasis may be potential causes. Lymphatic drainage obstruction is typically caused by congenital abnormal vessels or damage acquired to the lymphatic vessels from infection, trauma, surgery, or radiation. Several studies have reported that intralymphatic histiocytosis of the eyelid can occur with systemic diseases such as Melkersson-Rosenthal syndrome (MRS) or Morbihan disease.Information regarding intralymphatic histiocytosis is lacking in the ophthalmologic literature. Intralymphatic histiocytosis, first described by O’Grady et al, Morbihan disease often demonstrates as upper and bilateral facial lymphedema involving the eyelids with a history of rosacea. Only 8% to 25% of patients with MRS have a complete triad of orofacial edema, facial nerve palsy, and tongue furrowing. Morbihan disease, also known as rosaceous lymphedema or solid persistent facial edema, is an end-stage presentation of rosacea. The research on MRS and Morbihan disease includes reported cases in which patients manifest isolated unilateral eyelid swelling as a possible variant of these 2 diseases.MRS and Morbihan disease are in the spectrum of diseases associated with persistent facial lymphedema. These conditions, which are both accompanied by intralymphatic histiocytosis, share similar histopathological patterns. Despite the histopathological similarities of non-caseating granulomas, MRS and Morbihan disease often present with different clinical symptoms. While MRS often manifests as lower facial lymphedema involving the lips and tongue,Similarly, the patient in this study experienced unilateral periocular swelling for 7 years. Without any other histopathologic findings or additional triad features, we concluded that this was a type of intralymphatic histiocytosis. We consulted the dermatology department, which confirmed no evidence of rosacea. This case is similar to previous reports of monosymptomatic periocular MRS. However, all cases reported in Korea were accompanied by orofacial edema, facial nerve paralysis, or other symptoms and were managed without surgical intervention. To the best of our knowledge, this is the first reported case of intralymphatic histiocytosis with unilateral swelling in a patient of East Asian ethnicity with no history of any other inflammatory diseases. The patient underwent surgical debulking for biopsy and cosmetic improvement, followed by an intralesional triamcinolone injection.The histopathologic examination revealed perilymphatic, intralymphatic, and non-necrotizing granulomatous inflammation. The pathology inset of CD68 revealed that the intraluminal cells were diffuse histiocytes. The presence of CD31-positive endothelium surrounding the infiltrate confirmed their location within the lumen. Lymph stasis may cause inadequate clearance of antigens and localized immune dysfunction, which chronically stimulates histiocyte proliferation and aggregation. This can lead to chronic inflammation. We considered Langerhans cell histiocytosis or Rosai-Dorfman disease as differential diagnoses but ruled them out based on morphological and immunohistochemical findings. Immunohistochemically, the endothelial cells lining the dilated lumina exhibited CD31 immunoreactivity. The intraluminal histiocytes we observed did not exhibit emperipolesis or S-100 protein immunoreactivity. Additionally, CD1a showed reactivity. When the disease is associated with an underlying inflammatory condition or malignancy, the intralymphatic histiocytosis may resolve by treating the underlying disorder. Possible treatments include electron beam therapy, cyclophosphamide, topical and systemic steroids, amoxicillin and acetylsalicylic acid, pentoxifylline, NSAIDs, excision, pressure bandages, topical tacrolimus, and methotrexate. We observed partial symptom improvements after surgical debulking and a significant improvement in erythematous edema of the upper eyelid in this patient after intralesional triamcinolone injection. The unilateral eyelid edema responded to intralesional steroid injection. This case suggests that prioritizing surgical debulking followed by intralesional steroid injection can be an effective treatment for isolated eyelid edema, sparing other systemic treatments for recurrence.Diseases included in the spectrum of persistent facial lymphedema can be difficult to treat. Topical, intralesional, and systemic corticosteroids have been the mainstay of treatment, with varying results.Ophthalmologists encountering patients who suffer from persistent isolated eyelid swelling that does not respond to treatment should consider persistent lymphedema of the face. They should conduct a comprehensive history and physical examination to gather information about the patient’s history of rosacea, the location of the edema, and any other systemic diseases. Active communication with the dermatology and pathology departments can help facilitate a diagnosis. Although there are no specific guidelines for intralymphatic histiocytosis treatment, surgical debulking and intralesional steroid injection can be attempted.The authors would like to thank the patient for providing informed consent for publication.Conceptualization: Dong Hee Ha, Jeong Kyu Lee.Data curation: Dong Hee Ha, Jeong Kyu Lee.Investigation: Hee Sung Kim, Jeong Kyu Lee.Writing – original draft: Dong Hee Ha.Writing – review & editing: Hee Sung Kim, Jeong Kyu Lee.


Giant cell tumors (GCTs) of the ischium are rare and often diagnosed at an advanced stage. In fact, there is no defined treatment algorithm to treat this lesion. We present two case reports of Campanacci's stage three ischiopubic GCT confirmed with biopsy. They were effectively treated with excision of the ischiopubic ramus, aggressive curettage, drilling, and phenolization at the margins. The surgery was performed in a gynecological position with an approach over the ischiopubic ramus. Both cases present no recurrence (two and 10-year follow-up), and neither has a significant impact on the quality of life. A thorough plan and surgical technique were essential for the success of this intervention. Giant cell tumors (GCTs) of bone are locally aggressive benign neoplasms, although they can develop lung metastasis and suffer malignant transformation. They correspond to 5% of all primary bone tumors and 20% of benign tumors. Most cases are diagnosed between the second and fourth decades of life, with females being slightly more common. In 90% of cases, they appear in the epiphysis of long bones, close to the joint. About half of these tumors are found in the knee region  . The CamPelvic GCTs are very rare, corresponding to only 1.5%-6.1% of all GCTs . The claTherapeutic options include radiotherapy, intralesional resection, and wide resection, with or without adjuvant treatments such as radiotherapy, angioembolization, denosumab, and bisphosphonates .Pelvic GCTs present higher rates of complications and recurrence when compared to other sites . The ratThe best treatment should be adapted to the characteristics and anatomy of the lesion, namely the relationship with neighboring joints, as well as the patient's expectations. These lesions can reach considerable dimensions and extend into the acetabulum, leading to significant morbidity associated with the surgical treatment.Two patients, the first, a male in his 30s, and the second, a female in her 60s, presented with non-specific low back and sacroiliac pain and were diagnosed with Campanacci's stage three ischiopubic GCT, confirmed with biopsy Figure .The extension of the lesion to the posterior wall of the acetabulum in both cases precluded a wide resection. The first patient underwent preoperative angioembolization, and the second one completed one year of neoadjuvant denosumab Figure . ExcisioTreatmentSurgeries were performed with the patient supine in a gynecological position Figure .A longitudinal incision centered over the ischiopubic ramus was made Figure . After dIn the first case, the obturator nerve was sacrificed, and cement was applied to the acetabular margin to provide subchondral structural support.Histological examination of the resected lesion confirmed the diagnosis of a giant cell tumor of the bone in both cases.Outcomes and follow-up Both patients had uneventful wound healing.The first patient returned to work six months after surgery (as an intensive care nurse), however, he had paresthesias in the inner thigh and adductor weakness in the two months after surgery and developed chronic hip pain that was managed with oral pain medication. It has a mild impact on his daily activities. He had hip dysfunction and an osteoarthritis outcome score (HOOS) of 60.6% at 10 years of follow-up. He remains disease-free, without any signs of recurrence in the MRI Figure .The second patient is currently in the second year of follow-up and is asymptomatic after proper rehabilitation. She is disease-free from earlier breast cancer but presents slight peripheral neuropathy related to a previous systemic treatment (paclitaxel). She presents a HOOS of 86.9% and no signs of recurrence Figure .We report two clinical cases of ischiopubic ramus GCTs, locally aggressive with extension to the acetabular region, submitted to ischiopubic ramus resection and aggressive curettage at the margins.The first patient, in his 30s  , underweThe second patient, in her 60s, underwent neoadjuvant denosumab for one year with tumor size reduction and better delimitation. The sclerotic borders still allowed proper curettage after the ramus resection. Denosumab is a human monoclonal antibody effective in the treatment of GCT of bone that cannot be surgically removed or when severe morbidity is expected with surgical resection . The durThis specific anatomical location remains a challenge, and the adopted technique was successful. Surgical treatment of tumors affecting the acetabular region often results in functional impairment . The firDespite the challenge of the surgical approach, the complex anatomy of the pelvic region, and the local aggressiveness of GCT at the periacetabular area, we present two cases where the resection of the ischiopubic ramus and aggressive curettage of the margins were effective in treating and preventing recurrence with no significant morbidity to the patients.Due to its rarity and the complexity of the local anatomy, there is no consensus on the treatment of GCTs of the ischiopubic ramus. In order to define a treatment algorithm or protocol, more studies are required, preferably multicentric, to have a significant sample and reliable results.Ischium GCTs are rare lesions with significant recurrence rates in the first two years, especially when the lesion is not completely excised. It has an unspecific and indolent presentation, which leads to late diagnosis and treatment.An ischiopubic ramus resection is a good option for treating aggressive ischium GCTs. Extension of the tumor to the acetabulum may endanger the hip joint and preclude wide resection of the lesion, predicting a higher recurrence rate. Treatment must be individualized, depending on the stage of the disease, its location, and the expectations of the patient.