Pulmonary sclerosing pneumocytoma (PSP) is a rare lung tumor that is mostly isolated and commonly reported among middle-aged East Asian women. Recently, Immunohistochemistry (IHC) analysis has suggested that PSP is of primitive epithelial origin, most likely derived from type II alveolar air cells. Patients with PSP are generally asymptomatic and usually detected for other unrelated reasons during routine imaging. Several studies have already investigated the computed tomography (CT) features of PSP and their correlation with pathology. Magnetic resonance imaging (MRI) is a radiation-free imaging technique with important diagnostic value for specific pulmonary nodules. However, very few case reports or studies focus on the MRI findings of PSP.via thoracoscopic surgery. Histopathology and IHC results of the surgical specimen confirmed that it was a PSP. We concluded that the MRI findings of PSP might adequately reflect the different components within the tumor and aid clinicians in preoperative diagnosis and assessment. To the best of our knowledge, this is the most comprehensive case report on the MRI findings of PSP.We reported a case of an asymptomatic 56-year-old female with a solitary, well-defined soft-tissue mass in the lower lobe of the left lung. The mass showed iso-to-high signal intensity (SI) than muscle on T1-weighted image (T1WI) and T2-weighted image (T2WI) and a much higher SI on fat-suppressed T2WI, diffusion-weighted image, and apparent diffusion coefficient image. Contrast-enhanced fat-suppressed T1WI revealed noticeable inhomogeneous progressive enhancement throughout the mass. The mass revealed early enhancement without a significant peak, followed by a plateau pattern on dynamic contrast-enhanced MRI images. The patient underwent left basal segmentectomy The MRI findings of PSP correspond to its histopathological features. Here, we present a case of PSP with the most comprehensive MRI findings, emphasizing the importance of multiple-sequence MRI in diagnosing PSP. Pulmonary sclerosing pneumocytoma (PSP), formerly known as pulmonary sclerosing hemangioma, is a rare benign or low-grade malignant lung tumor that commonly occurs in middle-aged Asian women. It was first reported by Hubbell and Liebow  in 1955.A 56-year-old asymptomatic female was referred to The Fourth Hospital of Hebei Medical University  after a routine physical chest X-ray screening revealed a nodule in the left lower lung field. Physical examination showed no obvious positive signs. The patient had no history of surgery or trauma. There was no family history of hereditary diseases. The patient was presented with no respiratory symptoms and had no history of smoking or alcohol consumption. The patient was diagnosed with hypertension five years ago and has been maintaining normal blood pressure by taking medicine. There was no record of any past surgical procedures or traumatic incidents, and the patient had no family history of hereditary diseases. The results of blood tests and tumor marker analysis were within the normal range.The patient underwent pre-contrast and dual-phase  contrast-enhanced CT (CECT) scans of the chest five days before the operation. Pre-contrast CT image revealed a well-defined round soft-tissue mass in the lower lobe of the left lung, with a size of about 3 cm × 3 cm × 3 cm (2 and 800 s/mm2). Then, the patients underwent a pre-contrast volume interpolated breath-hold gradient-echo examination (VIBE), followed by the free-breathing dynamic contrast-enhanced (DCE)-VIBE sequence, started 20 seconds after a bolus injection of gadopentetate dimeglumine  at a dosage of 0.1 mmol/kg through a cubital vein at a rate of 3 mL/s by an MRI-compatible automated injector pump, followed by a saline chase of 20 ml at the same injection rate. The free-breathing DCE-VIBE sequence included 37 phases, with a total imaging time of 250 seconds.The MRI scan was performed with a 3.0 T MRI scanner  using a body phased-array coil (18 elements). The patient was scanned in the supine position throughout the examination. Pre-contrast MRI sequences included in-phase T1-weighted image (T1WI), out-phase T1WI, T2-weighted image (T2WI), fat-suppressed T2WI, diffusion-weighted imaging  than that of muscle and mixed with some low-SI foci within the mass .PSP was initially referred to as sclerosing hemangioma due to its presumed endothelial and vascular origin. Histologically, PSP is characterized by a distinct pattern of two types of epithelial cells , forming four architectural patterns: solid, papillary, sclerotic, and angiomatous, which vary in proportions , 5, 6. TIn the current case, the old female patient presented solely with a medical history of hypertension and did not exhibit any respiratory symptoms. The hypertension was appropriately managed, and blood pressure levels remained within the normal range. However, the relationship between hypertension and PSP remains unknown. CT is the primary imaging method for diagnosing PSP. Preoperative diagnosis of PSP based on CT findings is often challenging, especially when distinguishing it from other benign tumors, such as pulmonary hamartomas. MRI, as a functional imaging technique, may have the potential to provide additional diagnostic information on PSP. However, the final diagnosis of PSP still relies on the results of pathology and IHC results. The postoperative pathological results showed the tumor contained four characteristic histological patterns. Similar to previous studies , 9, the Typically, PSP presents as a single, slow-growing, well-circumscribed soft nodule on conventional CT images , 10. PSP-3mm2/s. This report is the first to present the value of DWI and ADC in diagnosing PSP, but the performance of other MRI sequences was not described. In our case, the tumor showed a high SI on the DWI image, with an ADC value of (2.36 ± 0.22)×10-3 mm2/s, indicating a benign tumor. PSP has the potential for growth despite its histopathological benign nature. Kim et al.  for the publication of any potentially identifiable images or data included in this article.YL, XG, NZ, and YW wrote the manuscript. HD undertook the pathological analysis of the specimens. HF, QW and GS undertook the radiological diagnosis. MW provided the clinical and surgical data of the patient. All authors contributed to the article and approved the submitted version.


Leiomyomas are the most common pelvic tumors, cervical uterine myoma being rare of all uterine fibroids with an incidence of 0.6% of all fibroids. Based on their location, cervical myomas can be classified as extra cervical  and intracervical. Cervical fibroids can further be anterior, posterior, lateral, and central depending on their position. The surgical treatment of cervical leiomyomas poses more difficulty; due to the risk of intraoperative Hemorrhage and the potential injuries because of contiguity and dislocation of adjacent organs. We present the case of a 46-year-old female, presenting with pain abdomen and abdominal distension. Contrast enhanced-magnetic resonance imaging showed a giant cervical myoma. Enucleation of myoma was done followed by total abdominal hysterectomy with bilateral salpingectomy. Injury to the ureter can be avoided with preoperative cystoscopy-guided bilateral ureteral stenting, intraoperative tracing of the ureter before applying a clamp, and dissection inside the fibroid capsule. The prevalence of uterine leiomyomas varies widely across literature, from 4.5% to 68.6%, depending on characteristics such as country/region, method of diagnosis, and health status . The uteA 46-year-old female, with two living issues, peri-menopausal, with previous two caesareans, last childbirth 16 years back, presented to the gynecology outpatient department with the complaint of pain abdomen and abdominal distension for two months. The patient complained of one episode of heavy menstrual bleeding in the last cycle. There were no altered bowel and bladder habits and no history of weight loss or appetite loss.General physical examination and systemic examination were normal except for mild pallor. The patient was class I obese with a body mass index of 34.2 kg/m2. On per abdominal examination, a large globular, firm mass with well-defined margins, non-tender with restricted mobility, corresponding to 28 weeks gravid uterus was palpable. The lower margin of the mass could not be made out. On per speculum examination, the cervix was flushed with the vagina. On bi-manual examination, a smooth, globular mass, up to 28 weeks in size of gravid uterus, firm in consistency with restricted mobility was felt. The uterus could not be felt separately and was bilateral adnexa free.A provisional diagnosis of large uterine fibroid was made based on clinical examination.Ultrasound findings- uterus was anteverted, with an endometrial thickness of 5.5 mm, a large heterogenous lesion in close relation to the uterus and right adnexa causing mass effect. Differential diagnosis includes sub-serosal fibroid, broad ligament fibroid, and multiple uterine fibroids.Contrast enhanced-magnetic resonance imaging Figure  showed aAfter pre-anesthetic workup, the patient was taken up for total abdominal hysterectomy with bilateral ureteric stenting. Her hemoglobin was 9.9 gm%, liver function test, renal function test, coagulation profile, and Papanicolaou test were within normal limits. On opening the abdomen, a giant fibroid of approximately 20 x 16 cm, was seen occupying the pelvis and abdominal cavity, uterus was sitting on the fibroid, giving the appearance of a Lantern on saint paul’s dome, as shown in Figure Dense adhesions were present between the peritoneum and anterior abdominal wall and with fibroid, which were separated. The uterus was infiltrated with vasopressin. Enucleation of fibroid with myoma screw was done after separating the bladder. Tracing of bilateral ureters was done and ensured to avoid injury. A total abdominal hysterectomy with bilateral salpingectomy was done. Intra-operatively two units of blood were transfused. Her post-operative period was uneventful. Bilateral ureteric stents were removed on post-operative day-21.Histopathological examination confirmed cervical fibroid with chronic cervicitis. Figure The cervical fibroids are rare and large cervical fibroids are rarer. Ferrari et al.  in theirSurgery either hysterectomy or myomectomy according to the patient’s age and childbearing remains the cornerstone in the treatment of cervical leiomyomas. Surgery can present difficulties, because of its position with the bladder anteriorly, rectum posteriorly, and bilateral ureters lying lateral to the cervix. In addition, the size of the fibroid can lead to alteration in the position of these structures and there is always a risk of intraoperative hemorrhage. In view of these facts, an experienced surgeon should perform surgery. Myomectomy can be performed via laparotomy, laparoscopy, robotic-assisted laparoscopy, or vaginally. Prevention of bleeding can be done using diluted vasopressin, bilateral uterine artery ligation, temporarily blocking uterine Artery flow with vessel clips, and the use of an Internal Iliac artery balloon occlusion catheter (IIABOC) . There iUreter injury remains a dreaded complication ,6. IntraA proper preoperative work up, knowledge of anatomy, and careful dissection of the bladder and ureters are required as most of the time anatomy is distorted. Injury to ureters can be avoided with preoperative cystoscopy guided. bilateral ureteric stenting, intraoperative tracing of the ureter before applying clamps, and dissection inside the fibroid capsule.


Pneumocephalus is defined as the presence of gas or air in the intracranial space and typically arises as a result of neurotrauma. Clinically, pneumocephalus most often presents asymptomatically but may cause headache, nausea, vomiting, and confusion. Pneumocephalus arising from mastoiditis is an unforeseen complication with only a handful of cases reported. We report a case of an elderly male who presented with stroke-like symptoms in the setting of erosive mastoiditis with pneumocephalus. Clostridium perfringens and Escherichia coli [Pneumocephalus involves air in the epidural, subdural, or subarachnoid space within the brain parenchyma or ventricular cavities . Common hia coli . Howeverhia coli ,5. Mastohia coli . This reAn elderly male with no past medical history, no antecedent trauma, and no symptoms prior to the current presentation for admission presented to our facility with new-onset, acute aphasia and dysarthria that began one hour prior to arrival. Initial neurological physical exam was significant for aphasia with no other focal neurological deficits including no motor deficits, no sensory deficits, and no cranial nerve deficits. On arrival, the National Institutes of Health Stroke Scale (NIHSS) score was 6, with deficits of failing to answer the month and age, severely fragmented language aphasia, and severe slurring of words with unintelligible speech. Initial computed tomography (CT) of the brain showed no evidence of an infarct, intracranial hemorrhage, or large vessel occlusion. Therefore, the patient was administered a systemic thrombolytic agent. Further review of the initial CT demonstrated acute bilateral erosive mastoiditis with an intracranial extension with pneumocephalus along the right tentorium cerebelli Figures , 2.The patient was admitted to the intensive care unit for the management of a presumed acute ischemic stroke and erosive mastoiditis with pneumocephalus. Broad-spectrum intravenous antibiotics were immediately initiated. As a management approach, otolaryngology and neurosurgery recommended a highly specialized procedure including bilateral mastoidectomies. As part of the cerebrovascular event timeline protocol, a magnetic resonance image (MRI) of the brain demonstrated layering purulent debris in the occipital horns of both lateral ventricles representing ventriculitis, given the presence of coalescing mastoiditis and no evidence of a dural sinus thrombosis, acute infarct, intracranial mass, mass effect, or midline shift Figures , 4.Clinically, the patient slowly improved with the resolution of focal neurological deficits after the initiation of broad-spectrum antibiotics with intravenous vancomycin and piperacillin-tazobactam. Therefore, he did not meet the criteria for an extracranial ventricular drain (EVD) with intrathecal antibiotics or lumbar puncture with cerebrospinal fluid analysis.However, after an extensive review of the brain imaging and clinical resolution of the neurological symptoms, it was concluded that the neurological presentation was highly due to the mastoiditis and ventriculitis with pneumocephalus than an acute cerebrovascular event or transient ischemic attack. After multidisciplinary discussion among infectious disease, neurology, neurosurgery, otolaryngology, and critical care, the decision was made to transfer the patient to a tertiary facility for further specialized bilateral mastoidectomies.Pneumocephalus occurs due to gas or air within the cranial cavity, with an overall low incidence rate of 0.5%-4.0% secondary to middle ear disease. It is most commonly associated with head and neck trauma, surgery, infection, and neoplasm . In the Although middle ear infections resulting in intracranial pathology only account for a small number of cases, 5%-26% result in mortality . SurgicaPneumocephalus is a rare complication of mastoiditis that can present with focal neurological deficits, deterring the main diagnosis. In the existing literature, there are a limited amount of cases describing such phenomena. A multidisciplinary approach to the evaluation and management is imminent to reduce mortality.


Mixed gonadal dysgenesis (MGD) is a rare sex development disorder, diagnosed by mosaic karyotype of 45,X/46,XY  with the presence of Müllerian structures, unilateral testis and contralateral streak gonad. MGD expresses diverse phenotypes, from female phenotype with virilization or turner stigmata, to ambiguous genitalia or male phenotype. Early diagnosis is crucial for effective correction of height, sexual development and cancer prevention. The study reports on a case of a 25-year-old patient, reared as female, presenting a large abdominal mass later confirmed as a mixed germ cell tumor. Associated findings were primary amenorrhea, ambiguous genitalia, short statue, gender dysphoria and hyperlipidemia. The study is the first to report on hyperlipidemia in MGD. Mixed gonadal dysgenesis (MGD) is characterized by mixed forms of defective gonadogenesis, resulting in unilateral testis, contralateral streak gonad and persistent Müllerian structures. Multiple cell lines can sporadically emerge via chromosome missegregation during early embryonic mitosis . Though diagnosis confirms 45,X/46,XY  mosaic karyotype , preciseA 25-year-old female with large abdominal mass and distension symptom was referred to our hospital’s gynecology clinic. Detailed history-taking revealed primary amenorrhea. She had an unremarkable antenatal and family history. According to her parents, her external genitalia (clitoris-like) were slightly large but unnoticed by physician at birth. She was diagnosed as phenotypic female and reared as a girl without further evaluation. There were no childhood illnesses, and her developmental milestones were average. In psychosexual development, at 8 years old, her gender identity and role were developed as a boy. At 12, she identified as a homosexual and was sexually active with a female partner. Her lack of concern for her absence of menstruation delayed her seeking medical consultation.2. Her arm span was 149 cm, and had no acne, nor deepening voice. Evaluation with Modified Ferriman–Gallwey score revealed mild hirsutism, with the score of 3 only at the upper lip area. She had no obvious Turner stigmata, nor dysmorphic features.In the physical examination, her height was 147 cm with a mid-parenteral height of 157.5 cm. She weighed 43.6 kg with a body mass index of 20.2 kg/mBreast development was Tanner stage I, while pubic hair Tanner stage IV. Abdominal exam found a 20-week-sized pelvic mass with firm consistency without tenderness. During the pelvic examination , a phallLaboratory findings revealed the hypergonadotropic hypogonadism state as following: FSH level 64.9 mIU/ml and an E2 level < 20 ng/dL. Thyroid-stimulating hormone and prolactin level were normal. The androgen panel was abnormally high for female range (2.76 ng/ml), while considered within the male range but with an abnormally low baseline (≤12 nmol/L [3.50 ng/ml]). LDL was 220 mg/dl, cholesterol 299.3 mg/dl, HDL 66.2 mg/dl and TG 94.4 mg/dl.Abnormally high level of three serum tumor markers was suspected of germ cell tumor: LDH 1740 U/L, AFP 5552 ng/ml, hCG 256 mIU/ml.Abdominal imaging revealed a large lobulated mass measuring 15.3 × 13.6 × 8.6 cm (suspected left-ovary in origin due to venous drainage to the left ovarian vein), a small uterus and the absence of right ovary with suspicious intra-abdominal testis (2.5 cm in diameter), at the right-side of the pelvis. No sign of invasion to adjacent organs or metastasis was reported. A transrectal ultrasound for uterine evaluation suggested a probable prepubertal-sized uterus (4 cm in length).In karyotype, 50 metaphases of peripheral blood lymphocytes were assessed with G-bands by trypsin, using Giemsa chromosome analysis, which resulted in 45,X [5]/ 46,XY[45].After multidisciplinary professionals’ evaluation, MGD with gonadal malignancy was suspected. Gynecologists and urologists prepared and operated an exploratory laparotomy for left gonadal tumor resection, a right gonadectomy and a hysterectomy. The surgical findings revealed a left gonadal tumor measuring 17.5 cm in diameter, with a tan-white smoothly lobulated surface and a mix of tan-white firm contents and fleshy necrotic tissue . SurgicaPostoperative systemic adjuvant chemotherapy, comprising bleomycin, etoposide and cisplatin (BEP regimen), was initiated promptly. After the fourth cycle of chemotherapy, her tumor markers were normalized, with no evidence of recurrence.With psychiatric and endocrinological support, the sex reassignment was concluded as male. The gender transformation was planned with post-treatment hormonal therapy.After completing the cancer treatment, gender reassignment and gender affirming hormone therapy were initiated following multidisciplinary team counseling and with the patient’s approval. Intramuscular testosterone replacement therapy was prescribed monthly. Lastly, although screening for other Turner syndrome’s comorbidities was normal, continuing surveillance was arranged.MGD is a complex sex chromosome abnormality, which genetically influences internal sex organ development. The phenotypic expression of anatomical and functional disturbances does not merely involve reproductive tract but multisystem. MGD is expected to cause gender dysphoria, gonadal tumors, developmental growth issues and currently is suspected to contribute to hyperlipidemia with atherosclerotic cardiovascular disease (ASCVD) risk. Due to adulthood presentation in our case, late outcomes were observed conclusively in four main aspects.Gonadal malignancy is reported to be highest in MGD among other disorder of sex differentiation (DSD), to which the presence of Y-chromosome is significantly related. Moreover, ambiguous phenotype in our case was found to be associated with higher carcinoma risk compared with mildly undervirilized male phenotype. This might be because tumors tend to be most pronounced in immature and/or poorly differentiated gonad. Thus, the degree of testicularization of the gonad is reflected by the expressed phenotype [henotype . Meanwhihenotype . Though Gender dysphoria: In individuals with 46 XY cell lines, the partial function of the Y-chromosome may contribute to the development of unilateral testis. Dysgenesis of Sertoli cells causes retention of Mullerian ducts and impairment of testicular androgenic function, resulting in incomplete masculinization of external genitalia and secondary sex characteristics. As MGD is usually diagnosed in childhood, a multidisciplinary team should manage sex assignment. Incongruity between assigned sex and experienced gender led to gender dysphoria in our case. Although gender dissatisfaction occurs in general population, it is more common in DSD patients. Predicting gender identity from karyotype, androgen exposure, genital virilization or assigned gender remains a challenge. The association between the rearrangement of Y-chromosome and gender is currently inconclusive [nclusive . In our Short stature is currently known to be related to 45,X cell by haploinsufficiency of the SHOX gene (one of the major growth regulating gene) [ng gene) . Despiteng gene) .Hyperlipidemia: Although multiple mechanisms underlie hyperlipidemia, sex chromosome and gonadal hormone may profoundly influence the development path leading to a lifelong consequence by gene expression. XO compared with XX women show increased body fat, whereas in 45,X Turner syndrome, dyslipidemia is prevalent since childhood [hildhood . Thus, thildhood , 10. GivThe unique outcomes in adult MGD with delayed diagnosis include not only gender dysphoria but also malignant gonadal tumor, growth failure and hyperlipidemia. A possible impact of sex chromosome in MGD on lipid metabolism was found. Due to the long-term ASCVD risk and its consequence on the overall health, intensive surveillance for dyslipidemia is recommended to enable appropriate preventive care for MGD patients.


An intramedullary abscess is an extremely rare form of infection of the spinal cord, with only about 100 cases reported in the literature. It typically presents with back pain, neurological deficits, and, occasionally, fever. The purpose of this article is to report a case of an intramedullary thoracic spinal cord abscess that was initially misdiagnosed as an intramedullary tumor.A 48-year-old female presented with chronic inter-scapular pain and lower limb weakness. The patient was initially misdiagnosed as a case of intramedullary spinal cord tumor, primarily due to the rarity of intramedullary spinal cord abscess (ISCA) as well as the absence of ring enhancement in the lesion on MRI. However, during a surgical procedure for the excision of the lesion, it was found to be a case of ISCA. The purulent contents of the lesion were evacuated, and the patient was treated with IV antibiotics, leaving the patient with a slight residual lower limb weakness on the follow-up examination.This case highlights the importance of considering intramedullary abscess as a possible diagnosis in patients with back pain and neurological deficits, even in the absence of ring enhancement on imaging. Prompt surgical intervention should be considered in probable cases of intramedullary spinal cord tumors. Intramedullary spinal cord abscess (ISCA) is a rare condition that can be easily mistaken for intramedullary tumors. The cervical location is the least commonly affected, except in cases of cryptogenic spread. The accurate diagnosis of ISCA relies heavily on the detailed examination of contrast-enhanced MRI images .ISCA can result in significant neurological damage and even death. The majority of cases arise from a direct source of infection, such as vertebral osteomyelitis or postsurgical infections. However, in cases where no source is found, the infection is known as "cryptogenic" and is associated with risk factors such as immunosuppression, intravenous drug use history, poorly controlled diabetes mellitus, or pulmonary arteriovenous malformations. In adults without these risk factors, cryptogenic ISCA is rare and can be easily misdiagnosed as more common conditions such as spinal intramedullary neoplasia, demyelinating disease, resolving hematoma, or infarction .A spinal cord tumor can be suspected based on contrast-enhanced MRI images, but surgery can reveal a spinal cord abscess. While spinal abscesses are occasionally seen in pediatric patients, intramedullary abscesses are extremely rare .An intramedullary spinal cord abscess is an uncommon infection of the spinal cord resulting in a purulent cystic lesion in the spinal cord substance. The common presentation is fever, pain, neurological deficits, and bladder/bowel dysfunction. The diagnosis is made through MRI, showing a hyperintensity on T2 diffusion-weighted imaging (DWI) and a hypointensity with a peripheral hyperintense rim on gadolinium post-contrast T1 DWI. Prompt diagnosis and management are necessary to avoid or limit permanent damage to the cord. The recommended course of treatment is a combination of surgical intervention for decompression and drainage and culture-specific antimicrobial therapy, though some cases have responded to antimicrobial therapy alone . Here, wA 48-year-old female presented to us with complaints of progressive bilateral lower limb weakness for the past five months leading up to the presentation and non-radiating midline inter-scapular pain for about three months that was intermittently exacerbated with physical activity. She had no history of any recent infection, immunocompromised state, immunosuppressant drugs, IV drug abuse, previous surgery, or trauma. Her postoperative HIV investigations came back negative. She denied experiencing any bowel or bladder dysfunction. On physical examination, she was found to be afebrile and had paraparesis with motor power of MRC  grade 2/5 in both lower limbs. All her sensory functions, including proprioception, touch, and vibration, were intact.The MRI scan of her spine showed a space-occupying lesion measuring 31 mm at the level of T4-T6 vertebrae, which was hyperintense on T2 diffusion-weighted magnetic resonance imaging (DW-MRI) and hypointense on gadolinium post-contrast T1 DW-MRI, but the latter did not show any peripheral ring enhancement as we would expect for an ISCA  for better control of the infection. After that, she was switched to oral antibiotics for the subsequent two weeks. During her course of treatment, she was found to be improving in terms of motor power in her lower limbs. An MRI scan of her dorsal spine showed a significant decrease in the size of the lesion. On her follow-up examination four months post-surgery, she was ambulating with only minor assistance, and her motor power was found to be 4/5 in both lower limbs. She was able to perform activities of daily living with minimal assistance, such as bathing, dressing, and feeding. She scored 2/6 on the Modified Rankin Scale.ISCA is an extremely rare entity, with only about 100 cases reported . The firISCA is characterized by a pus-filled cavity in the spinal cord that more often affects the cervical and lumbar cords and is relatively less common in the thoracic region, as in our case . The relIts occurrence is attributed to multiple risk factors, including an immunocompromised state like HIV infection and therapeutic immunosuppression, structural defects like dermal sinuses in the pediatric age group, previous infection, trauma, IV drug abuse, or previous surgery . The souA wide range of microbes have been isolated from intramedullary abscesses, with Mycobacterium tuberculosis being an important one in the developing world . Other cISCA is believed to present with a triad of fever, pain, and neurological deficits, though all these findings may not always be observed together in all patients . Other mThe diagnosis is made through history, clinical features, and magnetic resonance imaging. A ring-enhancing lesion on T1 DW-MRI is thought to be strongly suggestive of ISCA, but the absence of ring enhancement does not rule out ISCA, as we discovered in our case. The presence of the drop sign or precipitation sign  has also been reported, which, if present, may give a better clue to ISCA. A CSF examination may reveal elevated proteins, normal to low glucose levels, and leukocytosis .Concerning treatment, ISCA is best treated with a combination of surgery and antimicrobial therapy, though antibiotics alone may be effective in some cases . AntibioThe authors highlight the significance of considering ISCA as a potential diagnosis, despite its rarity, when faced with cases that resemble intramedullary tumors, particularly in instances where no ring enhancement is observed on T1 DW-MRI. Given the challenging nature of distinguishing between these conditions, the inclusion of ISCA in the differential diagnosis becomes crucial to ensuring appropriate and timely management. Failure to recognize and address ISCA promptly may lead to irreversible cord damage, which could result from an underlying, yet undetected, abscess. Therefore, the authors stress the importance of maintaining a high index of suspicion and employing thorough diagnostic evaluation to avoid overlooking ISCA and facilitate immediate surgical intervention when intramedullary tumors are suspected, thus mitigating the risk of potential long-term consequences.The authors strongly advocate for immediate surgical intervention in cases where intramedullary tumors are diagnosed to prevent the possibility of inadvertent harm to the spinal cord caused by a potentially concealed ISCA. Given the delicate and intricate nature of spinal cord pathologies, the urgency of accurate diagnosis and prompt treatment cannot be understated. The presentation of an ISCA without ring enhancement on T1 DW-MRI highlights the diagnostic challenges involved, making surgical exploration and intervention imperative to safeguard the patient's neurological function. By emphasizing the necessity for early surgical management in such cases, the authors aim to ensure that appropriate measures are taken swiftly to address the underlying pathology, mitigating the risk of irreversible cord damage and improving the overall prognosis for the patient.


In this case report, we present an extremely rare and previously unreported case of skull osteoma relapse without any attachment to the skull after hydroxyapatite cement (HAC) cranioplasty. The 49-year-old male patient was admitted with recurrence of the left frontal skull lesion; he underwent craniectomy and HAC cranioplasty for a left frontal osteoma 14 years before. Intraoperative findings disclosed multiple irregular lesions located on the HAC flap without any attachment to the bony structure and the roots of the lesions originating from the outer layer of the dura through several reserved holes. Pathological diagnosis was osteoma. The purpose of this report is to document this rare occurrence and provide the most probable pathogenesis for this rare event. They often occur on the long bones of the extremities, in the sinuses of the facial bones, in the skull, and in the mandible . Hydroxy22.1A 49-year-old male patient was admitted with recurrence of the left frontal skull mass; he underwent craniectomy and HAC cranioplasty for a left frontal skull osteoma 14 years before. Unfortunately, the clinical data from his first visit have been lost at the local hospital, and neither the patient nor his family know what material was used to repair the skull defect on the first surgery. Subsequently, three small hard masses could be touched under the scalp within the surgical area on the third year after surgery, and the masses could not be moved. There was no local tenderness and scalp swelling. The patient chose conservative treatment because of no obvious discomfort. However, the masses slowly grew up and affected the appearance gradually in the following 10 years. The physical examination revealed three distinct immovable masses protruding from the skin surface of the left forehead. They were hard in texture and smooth in edge, closely related to the so-called skull. The other physical examination and laboratory test (including colonoscopy) results were normal.CT scans showed partial defect of the left frontal skull with internal fixation shadow and multiple high-density nodules with a similar density as bone Figure 2The preoperative diagnosis of the osteoma recurrence was made based on the limited information provided by the patient. The operation was performed with adequate preparation and a lot of doubts, especially for how to reasonably interpret the preoperative imaging findings.2.2The intraoperative findings disclosed that the multiple irregular lesions were nodular and hard and showed obvious ossification, and they were on the HAC flap (2.3Pathology suggested the diagnosis of osteoma (3Osteomas are benign tumors consisting of mature normal osseous tissue, commonly encountered on long bones and in the mandible, sinuses of the facial bones, and the skull. The skull osteomas often have a good outcome without recurrence after surgical removal; however, they usually require cranioplasty , 4. HydrIn our report, the lack of information on repair materials makes it difficult for us to reasonably interpret the preoperative imaging findings; meanwhile, it also brings a lot of confusion for the preoperative diagnosis. Frankly, the preoperative diagnosis of the osteoma relapse was reluctant. Of course, the patient did receive craniectomy and HAC cranioplasty, which was from the intraoperative confirmation, due to frontal osteoma over a decade ago although the related data of the first surgery were lost. The intraoperative findings included the three-sided fixation of HAC and multiple lesions on the HAC without any attachment to the surrounding normal skull and the roots of lesions originating from the intact dura matter surface through several reserved holes. Honestly, these intraoperative findings were beyond what we thought the surgery would be. Multiple osteoma relapse without any attachment to the skull after HAC cranioplasty is extremely rare, which is firstly reported in the literature to our knowledge. In our case, the patient was highly suspected of relapsing on the third year after surgery according to his medical history description, but the patient chose conservative treatment without any discomfort. In the subsequent time period, the slow growth of osteomas with mild pathological characteristics might better explain why the relapse time was so long in this patient. More importantly, how to explain the pathogenesis more reasonably?In fact, the ossification of the roots on the intact dura surface was also suspected during operation, which was excluded by pathology results ultimately. Given the presence of multiple cranial osteomas, the patient underwent a colonoscopy, which revealed a negative result, to rule out Gardner syndrome . AlthougBesides, intracranial subdural osteomas without the involvement of bony structure are rarely reported, with only approximately 20 cases having been reported to date, and the mechanism behind their genesis remains unclear , 7, 8. HThe case leaves us with many doubts, including whether the HAC holes are just a coincidence to promote their growth and whether the osteomas will relapse again during the long follow-up because there was no special treatment, just cauterization, for the intact dura at the second surgery.4In this case report, we present an extremely rare and previously unreported case of skull osteoma relapse without any attachment to the skull after HAC cranioplasty. The pathogenesis of osteomas without bone involvement is prone to the osteoblasts of osteoma implantation during the first surgery.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving human participants were reviewed and approved by the Ethics Review Board of the West China Hospital of Sichuan University. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the patient for the publication of this case report.All authors contributed to the diagnosis and treatment of the patient. XY performed image data analyses and wrote the manuscript. YL contributed to the operation and prepared the manuscript. YZ helped perform the analysis through constructive discussion. All authors have contributed to the manuscript and approved the submitted version.


The diagnosis of retroperitoneal sarcoma may be challenging for the radiologist. Current guidelines report postsurgical margin as the strongest predictive factor for disease-specific survival and recurrence, as well as histologic subtype and grade. The role of the radiologist is indeed important in RPS diagnosis, management, and follow-up, as the ability to promptly recognize local progression, invasion of nearby structures, and complications has a direct impact on patients’ survival. A practical guide is provided to radiologists with an overview of the current knowledge regarding cross-sectional CT/MRI imaging features of patients with retroperitoneal sarcomas, presenting tips and tricks to improve imaging diagnosis of RPS.Primary retroperitoneal sarcomas (RPS) represent around 10–16% of all sarcomas, with liposarcomas and leiomyosarcomas being the most common subtypes. RPS have some peculiar characteristics, imaging appearances, worse prognosis, and complications compared to other locations of sarcoma. Commonly, RPS primarily present as large masses, progressively encasing adjacent structures, causing mass effect, and complications. RPS diagnosis is often challenging, and these tumors may be overlooked; however, failure to recognize RPS characteristics leads to a worse prognosis for the patients. Surgery is the only recognized curative treatment, but the anatomical constraints of the retroperitoneum limit the ability to achieve wide resection margins; therefore, these tumors have a high rate of recurrence, and require long-term follow-up. The radiologist has an important role in the diagnosis of RPS, the definition of their extent, and their follow-up. Specific knowledge of the main imaging findings is required to reach an early diagnosis, and, ultimately, to guarantee the best patient management. This article provides an overview of the current knowledge regarding cross-sectional imaging features of patients with retroperitoneal sarcomas, presenting tips and tricks to improve imaging diagnosis of RPS. Retroperitoneal sarcomas (RPS) are rare and aggressive tumors, with an incidence of 0.5–1 new case per 100,000 inhabitants per year ,3, accouThe retroperitoneum is a particular location for sarcomas. Anatomic constraints limit the ability to achieve wide resection margins ,2, and sIn several series, recurrence rates ranged from 45 to 49% within the first five years and from 60 to 82% at 10 years after initial treatment ,15, follCurrent guidelines report postsurgical margin as the strongest predictive factor for disease-specific survival and recurrence, as well as histologic subtype and grade ,18; howeIn RPS, as well as in other sarcomas, lymph node metastases are rare (<1%) . A combiLastly, it is important to refer patients with suspected RPS to specialized tertiary referral centers ,3,18. BlThe role of the radiologist is indeed important in RPS diagnosis, management, and follow-up, as the ability to promptly recognize local progression, invasion of nearby structures, and complications has a direct impact on patients’ survival. The rarity and diversity of RPS initial clinical and radiological presentations make them challenging to diagnose. The aim of this review is to present the current state of the art on RPS diagnosis, treatment, and follow-up, presenting the main radiological findings connected with principal RPS subtypes on cross-sectional imaging, providing tips and tricks to help reach an early and correct diagnosis.The detection of retroperitoneal masses includes a wide spectrum of differential diagnoses that must be ruled out, such as metastatic adenocarcinoma, retroperitoneal fibrosis, lymphoma, germ cell tumor, paragangliomas, or Castleman’s disease . The diaContrast-enhanced computed tomography (CT) is the most useful and widely available first-line imaging technique ,3,4,5,6.Contrast-enhanced CT of the chest is indicated by the guidelines . Brain MFollowing appropriate imaging assessment, image-guided percutaneous coaxial core needle biopsy is needed to confirm the diagnosis ,4,11,18.Recurrence on imaging may antedate symptomatic recurrence by months to years. Postsurgical imaging follow-up should be performed every 3–6 months for the first 2–3 years, and then annually after five years .With regards to imaging protocols, multiphasic study is recommended, including arterial, portal venous, and delayed phase on CT to better assess local extension, invasion of nearby structures, metastases, or local complications. The relationship with major vessels is a key element in radiological evaluation, as it affects surgery, neoadjuvant treatments, resection, and bypass reconstruction . Sambri Contrast-enhanced MRI study is aimed at tissue characterization. Imaging protocols should include T2- and T1-weighted (w) sequences, on axial and coronal planes, with and without fat suppression, to assess the presence of blood, fluid, gross fat, and fibrotic tissue. DWI and ADC maps are important to depict local and distant extension, and T1-w Dual Echo sequence to assess microscopic fat. Recent studies are exploring the promising role of radiomics in preoperative imaging for pathological prediction , assess All sarcomas on CT have typically a heterogeneous appearance. Sarcoma may be slightly vascularized or early enhancing and can contain necrotic or fluid areas ,34. ThisLPS can contain large quantities of gross fat, thus making their extent assessment very difficult ,33,34. TRadiologists should describe tumor location, size in the three-dimensional planes, the relationships and mass effect to other structures and vessels, parietal wall, nervous and vascular structures, and spine, as well as the presence of fat, myxoid, necrotic, and hemorrhagic content ,33. RegaA.Well-differentiated liposarcomas  are low-LPS represent the most common primary RPS ,13,14,34B.Myxoid/Round-cell liposarcomas (MLS) are intermediate-grade tumors and almost always occur in the retroperitoneum as secondary locations . They arC.Dedifferentiated liposarcomas  are highD.Pleomorphic liposarcomas contain little or no fat and myxoid components. They are considered high-grade malignancies with high rates of local recurrence and distant metastases . They arE.Undifferentiated pleomorphic liposarcoma: imaging features are nonspecific. It manifests as a large, well-circumscribed soft-tissue mass with heterogeneous enhancement and myxoid components. Areas of necrosis and hemorrhage may be seen but are less extensive than leiomyosarcomas. Calcifications occur in up to 20% of cases with a ring-like pattern. Direct invasion of adjacent organs may be present .The main differential diagnosis is with simple lipomas  that wilAfter surgery, retroperitoneal LPS have a high tendency to recur locally, while metastases are less common. Intraoperatively, low-grade LPS is grossly like normal fat, thus making the resection challenging, as frozen section evaluation is considered unhelpful . PreoperLMS is a smooth muscle tumor , more coAlthough their imaging appearance is nonspecific, most solitary fibrous tumors  show hetIn the retroperitoneum, a mass originating proximally to the spine, in the psoas muscle, in the sciatic nerve, or sacral plexus, or involving the neural foramina (which must be checked) can suggest a neural origin ,47. SchwBenign NST are usually rounded and well defined, with subtle, homogeneous enhancement, but the differential diagnosis with MPNST is oftentimes not doable on imaging ,40. The Some CT features can help to identify the organ of origin of RPS:The beak (claw) sign: when a mass deforms the edge of an adjacent organ into a “beak” shape, it is likely that the mass arises from that organ ,46)11,46The embedded organ sign: when there is intimate contact between the mass and the organ of origin, a desmoplastic reaction and sometimes ulcerations are observed (positive sign). On the contrary, a moldable, compressed organ will be deformed into a crescent shape (negative sign). The latter is useful for example in the differential diagnosis between RP leiomyosarcoma and primitive inferior cava leiomyosarcoma ,46.The phantom or invisible organ sign: if an organ cannot be seen, it is probably the origin of the mass. This sign can lead to false positives diagnoses, as large sarcomas can invade the adrenal gland.Prominent feeding artery sign: hypervascular masses in most cases are supplied by feeding prominent arterial vessel that can be studied on CT and can help to understand the origin of the mass .The sandwich (or hamburger) sign : presencCT is also useful to identify patterns of growth and dissemination, which is fundamental for surgical and treatment planning in multidisciplinary contexts, and useful to identify the sarcoma subtype. Lesions surrounding normal structures and vessels without invading or compressing them and affecting more than one compartment will more likely be lymphangiomas and lymphomas; conversely, lesions that are located along the sympathetic chain will most likely be paragangliomas or ganglioneuromas.Absence of enhancement is commonly seen in cysts, hematomas, and simple lipomas.Early enhancement and rapid washout is seen in paragangliomas and Castleman’s disease.Early enhancement and delayed or imperceptible washout is unspecific, as it is seen in most tumors , such as paragangliomas.Late enhancement is commonly seen in neurogenic tumors, desmoids, leiomyomas, leiomyosarcoma, and lymphoma.The assessment of the enhancement pattern is another fundamental aspect to search on CT to understand the nature of a lesion, as follows:Lastly, CT can also help to identify tumoral components such as fat, myxoid stroma, necrosis, cystic degeneration, and calcifications. Calcifications can be present and indicate dedifferentiation and poor prognosis or sclerosing or inflammatory variants of well-differentiated LPS .Contrast-enhanced MRI is the best modality to characterize a soft-tissue tumor, but also for local staging and prognosis. The diagnostic orientation can be facilitated on MRI by the identification of myxoid stroma , fatty content  and has been associated with an increased risk of local recurrence and worse prognosis , as it iIn LMS, instead, a prognostic factor is the quantification of necrosis, as higher necrosis is associated with higher grade of the disease .In all RPS, evidence of osseous invasion is linked to lower disease-free survival and overall survival, while vascular or nerve invasion and multifocality are associated with higher local recurrence rates. Nodal invasion instead correlates also with higher probability of metastases ,35.MRI helps also in the differential diagnosis with lymphoma facing a hypointense T1 homogeneous mass with a homogeneous enhancement and strong diffusion restriction, without necrotic, blood, fibrotic or fat signal, no peritumoral enhancement or abnormal intra- and peritumoral vasculature, and a possible infiltrative growth pattern .Complications are usually related to tumor invasion of adjacent structures or to mass effect caused by the tumor and include hydronephrosis, pulmonary embolisms secondary to compression of inferior cava vein, nerve compression, bowel obstruction  49], an, an49], An optimal management of RPS is facilitated by a comprehensive pretreatment diagnosis and staging, for which the radiologist has a primary role. Pretreatment pathologic diagnosis is needed, but complete surgical resection with negative margins is the cornerstone for nonmetastatic RPS and is the only proved curative strategy. This is achieved by en bloc resection of the tumor and all surrounding structures, even if not overtly infiltrated on imaging or surgical exploration ,17,18,51In cases of recurrence, surgery should be aimed at achieving macroscopic complete resection, including surrounding organs only when overtly infiltrated . GrosslyCurrently, there is no evidence to support the routine use of adjuvant radiation therapy (RT) or adjuvant chemotherapy in RPS . NeoadjuRadiologists play a primary role in preoperative diagnosis and staging of RPS, detection of recurrence, and assessment of postoperative complications. Many imaging patterns can overlap among the various subtypes of sarcomas. Nonetheless, some specific imaging appearances can guide the radiologist. This is of the utmost importance, as imaging has a central place in each phase of the management of patients, and both nonspecialized and specialized radiologists can have a strong impact in the management and survival of patients with RPS. Indeed, the radiologist is commonly among the first physicians to raise the suspicion of the diagnosis of sarcoma. Imaging features with patterns of involvement, associated to clinical features and a multidisciplinary approach through discussions in tumor board, is fundamental.


Lymphangiomas are benign malformations of the lymphatic vessels which can be primary or secondary in etiology. Colonic involvement is rare, and the diagnosis is mostly incidental. Sometimes, the initial endoscopic appearance can be misleading. We present a case of colonic lymphangiomatosis presenting with free air under the diaphragm requiring surgical removal of the involved portion of the colon. The diagnosis was confirmed by the pathology of the resected specimen and its correlation with prior clinical information. The patient recovered well with an uneventful postoperative course and follow-up. This case demonstrates a rare complication of colonic lymphangiomatosis prompting definitive treatment by surgical resection. Multiple colonic lymphangiomas, or colonic lymphangiomatosis (CL), are a rare clinical entity and are mostly diagnosed incidentally . With thA 73-year-old male with a history of diabetes mellitus and chronic kidney disease underwent computed tomography (CT) abdomen and pelvis with IV contrast for the evaluation of microscopic hematuria of nearly three months duration, which showed the incidental finding of free intraperitoneal air. At this time, the patient did not have abdominal pain, fever, or any signs of sepsis. The patient had a history of colonoscopy for colon cancer screening a few weeks prior to this presentation which showed a "large concentration of polyps" in the sigmoid colon, not amenable for endoscopic removal. The patient had plans for a sigmoidectomy in the coming weeks. However, nearly four weeks after the initial CT scan, the patient developed abdominal pain and nausea, and labs showed worsening leukocytosis 17 K/µL (reference range: 4.0-10.5 K/µL) from the previous level of 9.9 K/µL three months ago. Repeat CT abdomen at this time revealed moderate free intraperitoneal air, worse as compared to previous, and innumerable colonic mucosal cysts of the very redundant sigmoid colon. The patient underwent exploratory laparotomy, and although no frank overt perforation was seen, the sigmoid colon exhibited soft-tissue fullness during the gross intraoperative examination. Given the planned sigmoidectomy given the prior endoscopic findings, a partial sigmoid colectomy with primary anastomosis was performed.The resected specimen showed large contiguous well-circumscribed and simple cystic structures (up to 3 cm in size) giving a sessile “polypoid” appearance of the mucosa Figure . HistoloLymphangiomas are rare benign tumors that are mostly incidentally found on colonoscopy, and most of them do not require resection . As per Besides colonoscopy, imaging in the form of an abdominal CT scan, barium enemas, or endoscopic ultrasound has been used for the diagnosis ,6. BesidIn conclusion, CL should be considered as a differential in patients with multiple aggregated mucosal lesions that appear like polyps on endoscopy. Clinicians must have a high suspicion especially if such lesions are noted to be aggregated in a specific area of the colon. Free intraperitoneal air may suggest impending clinical decline in such patients which can be prevented by prompt diagnosis and clinical management.


Contrast defects in veins are often diagnosed as benign thrombi, but depending on the patient’s background it is necessary to differentiate between tumor thrombi. It is difficult to differentiate between these using contrast‐enhanced CT alone, but with angioscopy it is easy to visually distinguish between a benign and tumor thrombi.Contrast‐enhanced computer tomography (CT) performed on a male patient being treated for de‐differentiated chondrosarcoma revealed contrast defects in the pulmonary artery and right femoral vein, and a diagnosis of pulmonary artery thromboembolism and venous thromboembolism was made, and oral anticoagulant therapy was started. However, a follow‐up CT showed that the contrast defect had extended to the inferior vena cava. Observation using an angioscope revealed that it was not a benign thrombi but a tumor. A contrast defect was observed in inferior vena cava (IVC) of a 61‐year‐old male patient with de‐differentiated chondrosarcoma, but it was not possible to distinguish whether it was a thrombus or a tumor. Using an angioscope, he was able to clearly diagnose a tumor. A 61‐year‐old Japanese man visited orthopedics complaining of pain in the right thigh and was diagnosed with dedifferentiated chondrosarcoma (DCS) arising in the right femur after needle biopsy and surgical tumor removal. Preoperative contrast‐enhanced computed tomography (CT) revealed a contrast defect in the right femoral vein Figure . We suspPrevious reports have demonstrated that FDG‐PET and T2‐weighted MRI can distinguish benign and tumor thrombi.Shiho Wakasa: Writing – original draft. Hiroya Hayashi: Investigation; methodology; writing – original draft. Takanori Yamazaki: Conceptualization; funding acquisition; methodology; project administration; visualization; writing – review and editing. Yasuhiro Izumiya: Writing – review and editing. Daiju Fukuda: Writing – review and editing.This work was supported by a grants‐in‐aid of The Cardiovascular Research Fund, Tokyo, Japan.The authors declare that they have no conflict of interests.This case study was conducted according to the principles of the Declaration of Helsinki and Ethical Guidelines for Medical and Health Research Involving Human Subjects by the Ministry of Health, Labor, and Welfare and Ministry of Education, Culture, Sports, Science, and Technology of Japan. This case study was approved by the Ethics Committee of Osaka Metropolitan University .Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.Is required for your own works published by other publishers and for which you did not retain copyright.None.Video S1.Click here for additional data file.


Patients complaining of rectal bleeding, constipation, and a suspicious mass in colonoscopy should undergo biopsy. Histological features such as fibromuscular obliteration in the lamina propria favor SRUS, a benign disorder. Solitary rectal ulcer syndrome (SRUS) is a rare benign rectal disorder often misdiagnosed with other diseases, such as IBD, constipation, and malignancies. Therefore, it is necessary to know its diagnostic features and treatment. In this report, we present a case of a polypoid solitary rectal ulcer presented with rectal bleeding and chronic constipation mimicking rectal neoplasm on colonoscopy. Histopathological study showed the characteristics of SRUS, the most important of which was fibromuscular obliteration in the lamina propria. Therefore, it is necessary to know its diagnostic features and treatment.This report presents a case of a polypoid solitary rectal ulcer in a 19‐year‐old man who came to the gastroenterology clinic with rectal bleeding and chronic constipation, the diagnostic procedures to rule out other critical differential diagnoses, and the treatments performed.2A 19‐year‐old man came to the gastroenterology outpatient clinic complaining of rectal bleeding and severe constipation. He had a history of constipation and tenesmus since childhood. His symptoms intensified, and presented with rectal bleeding recently, so he came to the clinic. He did not have any past medical or past surgical history and did not use any medication. His family history of bowel disease and colorectal cancers were unremarkable. He had no complaint of abdominal pain or recent weight loss.At First, general physical examinations were performed, and there were no abnormalities except pallor. Digital rectal examination was impossible due to partial obstruction of the rectal lumen. His blood pressure was 115/75 mm Hg; his heart rate was 96 bpm; his body temperature was 36.6°C; his respiratory rate was 18; and his oxygen saturation was 97% without supplementation.On his laboratory investigation, his white blood cell count was 7600; his hemoglobin was 10.9 gm/dl; his hematocrit was 38.8%; his mean corpuscular volume was 65 fl; his ferritin was 6.3 ng/mL; his platelet count was 361,000; and his erythrocyte sedimentation rate was 5 mm/h. These findings indicated hypochromic microcytic anemia, probably due to iron deficiency anemia. Coagulation profile and liver function tests were within normal ranges. Examinations of stool for parasites and cultures were negative.Colonoscopy was performed according to the indications in this patient, such as rectal bleeding and chronic constipation. It revealed one large semi‐circumferential infiltrative mass lesion with a polypoid appearance in the rectum within 5 cm of the anal verge, which was suspected to be malignant, so biopsy was taken Figure . The entHistopathological study revealed features of solitary rectal ulcer, including fibrosis and extension of smooth muscle fibers in the lamina propria with diamond‐shaped crypts, crypt regenerative hyperplasia, surface erosion, crypt hyperplasia, ecstatic capillaries, and minimal inflammation Figure . These hTreatment with bulking agents, hydrocortisone, and mesalamine enemas was prescribed, and he was advised to have a high‐fiber diet. During the follow‐up visits, his symptoms, such as constipation and hematochezia, were alleviated. Now he is under the care of a gastroenterologist and advised to present regularly to perform follow‐up with colonoscopy examination.3SRUS is a spectrum of clinicopathological abnormalitiesThe most common symptoms of SRUS are rectal bleeding, constipation, anemia, and abdominal pain.The polypoid appearance of this lesion may be misdiagnosed with an inflammatory polyp, hyperplastic polyp, or rectal carcinoma leading to delayed diagnosis and treatment, which can cause several complications, so it is necessary to have biopsies for a definite diagnosis.In our case, histologic features were fibrosis and extension of smooth muscle fibers in the lamina propria with diamond‐shaped crypts, crypt regenerative hyperplasia, surface erosion, and minimal inflammation. In order to distinguish SRUS from dysplasia, minimal inflammation and crypt regenerative hyperplasia are important features. However, crypt regenerative hyperplasia along with extension of smooth muscle fibers in the lamina propria around the crypts, may lead to a misdiagnosis of invasive adenocarcinoma, so an expert gastrointestinal pathologist should be consulted to confirm the diagnosis.In order to distinguish SRUS from IBD, there are two important histologic features, the presence of smooth muscle between crypts and ulceration without the infiltration of chronic inflammatory cells. Furthermore, the most critical differential diagnosis is rectal carcinoma; which may have similar appearance to SRUS on colonoscopy, such as thickened mucosa and strictures; so, histological findings are critical, especially in patients over 40 years of age. The differential features of SRUS from rectal cancer are summarized in Table Treatment is based on the severity of the disease and the patient's symptoms, such as the severity of bleeding, constipation, ulcers, and rectal prolapse. Patient education and lifestyle changes are the first steps of treatment.In conclusion, for patients with complaints of rectal bleeding with constipation and the presence of a suspicious mass in colonoscopy, a biopsy should be taken to decide on a treatment plan, because it differs based on the nature of the mass. Histological features such as fibromuscular obliteration in the lamina propria is in favor of SRUS; however, IBD and malignancies should be ruled out.Masood Faghih Dinevari: Conceptualization; methodology; project administration; supervision. Amirtaher Eftekharsadat: Conceptualization; methodology; supervision. Mahdi Tarverdizadeh: Data curation; investigation; validation. Seyyed Mahdi Rasulimanesh: Data curation; investigation; validation. Ali Riazi: Data curation; validation; writing – original draft. Amirreza Jabbaripour Sarmadian: Writing – original draft; writing – review and editing. Samaneh Abbasian: Data curation; project administration; writing – original draft; writing – review and editing.There was no financial support or funding for this research.The authors declare no financial and non‐financial competing interests related to this work.This study was performed according to the principles outlined by the World Medical Association's Declaration of Helsinki on experimentation involving human subjects, as revised in 2000, and was approved by the Tabriz University of Medical Sciences ethics committee with the approval number IR.TBZMED.REC.1399.1101 on 2021/02/22.The patient was informed regarding publishing this case report, and written informed consent was obtained to publish this report under the journal's patient consent policy.


Alveolar adenoma is a rare benign tumour, usually presenting as a peripherally located solid mass, sometimes mimicking malignancy.A 37-year-old woman presented with chronic intermittent vague chest discomfort. The chest x-ray showed a simple cyst in the left lower lung field, and serial computed tomography (CT) over the following 2-year period showed rapid growth of the cyst, from 3.5 to 9.0 cm in diameter. The CT scan suggested bronchiolar communication, which was suspected to be the cause of growth, via check-valve mechanism. Thoracoscopic surgery was performed, and we found a thin-walled cyst in the lingular segment. Wedge resection was performed and the pathology was an unexpected alveolar adenoma which had grown on the terminal bronchiole, causing the alveolus to rupture and the cyst to grow. In 48 months of follow-up, there was no evidence of recurrence and the patient’s symptoms resolved.Rapidly growing pulmonary cysts can lead to complications including rupture with pneumothorax and haemothorax, and surgery is always indicated. Abnormally rapid growth may indicate an underlying pathology such as alveolar adenoma. Surgical resection is the treatment of choice and there have been no reported cases of recurrence. Here we present a rare form of alveolar adenoma, which was a form of rapidly growing pulmonary cyst. Small pulmonary bullae are a common incidental finding, but large pulmonary cysts are rare and are usually discovered incidentally during routine check-ups or through imaging (chest x-ray or computed tomography [CT]) acquired for other reasons. Whether or not surgical resection is necessary for large pulmonary cysts depends on the number and characteristics of the cysts, the underlying pulmonary disease, and the presumed cause of the cysts.The differential diagnosis includes congenital lung disease , infectious disease, autoimmune disease, and malignancy . AlthougA 37-year-old woman was referred to our hospital after a health check-up at another hospital. She had an approximately 2-year history of vague chest discomfort and heaviness, and the chest x-ray from the referring hospital showed a cystic lesion in the left lower lung field. Abdominal ultrasound and routine blood tests results from the other hospital were all normal. She denied ever smoking and had no known comorbidities. Complete blood count with differential, blood chemistry, and electrocardiogram (ECG) were reviewed, and the results were all within normal limits.The cyst diameter on the initial chest x-ray was approximately 3 cm. The baseline low-dose chest CT showed a 35-mm intraparenchymal pulmonary cyst of the lingular segment Fig. A. After After general anaesthesia with double endotracheal intubation, the patient was placed on one-lung ventilation. Two-port video-assisted thoracoscopic surgery (VATS) was performed. On intraoperative inspection the thin-walled, broad-based cyst measured approximately 9 cm and was located in the lingular segment adjacent to the fissure Fig. D and E. On gross examination, the lesion was described as a unilocular cavity with an irregular wall with a maximum thickness of approximately 2 mm marked by fibrous thickening in the vicinity of the pleura  drain on postoperative day 1, and the patient was discharged on postoperative day 3. Given the rare but benign nature of the lesion, a brief multidisciplinary consultation with the pulmonologist, pathologist, and radiologist was called but no specific additional treatment besides regular follow-up was recommended. The patient remained symptom-free after surgery and there was no evidence of recurrence during a 2-year follow-up period , followed by the right upper lobe (15 cases), left upper lobe (9 cases), and right upper lobe (8 cases). The mean size of the tumour is 2.5 cm, ranging from 0.2 to 9.1 cm. The alveolar adenoma size itself in the present case was also 2 mm, although the total size of cyst was 9 cm. The CT findings were mostly of solid, round, well-circumscribed, homogeneous lesions of the peripheral lung; purely cystic features, as in our case, were reported in 2 cases, and solid lesions with cystic appearance in 4 cases.According to Fujimoto et al., magnetic resonance imaging (MRI) may be productive in some cases for delineating cystic spaces with central fluid and thin-rim enhancement . MRI is Surgical resection has been performed in most cases for both diagnostic and therapeutic purposes after CT scan. Fluorine-18-fluorodeoxyglucose positron emission tomography (PET) has been performed in only a few cases, and in most of these there was no evidence of infection or malignancy in the suspicious nodule. Roshkovan et al. reported an SUVmax of 0.6 for a 12-mm solid nodule, which was only slightly above the lung parenchymal background . NosottiThe pathogenesis of such tumours remains unclear, but their origins from type II pneumocytes is the consensus view. There is indeed some evidence supporting a transition to type II pneumocytes by primitive mesenchymal cell precursors , 11. MicThere have been other cases where tumour growth has occurred during follow-up before surgery , 24. AltCyst formation is a well-known characteristic of some types of malignant tumours. In squamous cell carcinoma, the cavitation is related to the tumour necrosis and keratinization. In adenocarcinomas, cavitation can be attributed to the tumour necrosis or to a unidirectional check-valve mechanism , 26. As Because the clinical presentation is so imprecise, a diagnosis of alveolar adenoma is challenging to validate. Frozen sections or small biopsy specimens may closely resemble normal lung tissue . At timeEfforts to distinguish alveolar adenoma are especially critical when presenting as a solitary peripheral nodule. The decision for surgery is made chiefly for diagnostic purposes. Progressive growth thus warrants surgical resection for tumour verification, ruling out malignancy , 30. At It is interesting to note that although the exact relationship is not clear, there have been some cases of co-existence of alveolar adenoma with malignant diseases or other benign adenomas. Alveolar adenomas have been found incidentally in several patients during the evaluation of other conditions including as prostate carcinoma, colorectal carcinoma, and pituitary carcinoma , 22, 31.Many of the points above are exactly why we need a multidisciplinary discussion. As well described in the interdisciplinary paper by Roshkovan et al., there are a number of questions to be answered. Among these are: should biopsy be performed before surgery? Is systematic workup, including PET, necessary before surgery? How much surveillance is necessary before surgery? What surgery should be decided and how extensive should surgery be? And what type of postoperative follow-up is required? . In thisIn conclusion, alveolar adenoma is rare and considered benign. However, with the potential for gradual progression and a report of malignant transformation, the treatment of choice is complete surgical resection. No recurrences have been reported after curative surgery.


Streptococcus bovis. He was treated with cephalexin (22 mg/kg PO BID) for 30 days. At the time of his one-month recheck, he was swallowing normally with no neurologic deficits. He remains normal at the time of this report (17 months later). This case reports a successful explant of a chronic atlantoaxial implant infection.An 11-year-old male neutered Yorkshire Terrier was presented with a cervical mass that developed a draining tract. Aside from the dysphagia reported by the owner, his neurologic exam was normal. Three years prior, the patient was diagnosed with an atlantoaxial subluxation that was ventrally stabilized with polymethylmethacrylate (PMMA) and self-tapping titanium screws. There were no postoperative complications until presenting with the cervical mass and dysphagia. Computerized tomography (CT) of the cervical spine confirmed caudal migration of the PMMA and screws with an abscess surrounding the implant. A surgical explant of the PMMA and screws was performed without complication. The atlantoaxial joint remained normally aligned on postoperative radiographs. Cultures of the implant grew  Atlantoaxial instability (AAI) is a well-recognized problem in primarily young, toy-breed dogs in veterinary medicine , 2. CommAlthough medical management is an option for treatment of AAI, various surgical techniques have been developed over the past several years to find the ideal approach to reduce postoperative complications –7. VentrAn 8-year-old male neutered Yorkshire Terrier presented to the University of Florida in May 2018 for an acute onset of cervical pain and ataxia. On neurologic exam, the dog was ambulatory with tetraparesis and cerebellovestibular ataxia. There were delayed to absent postural reactions on all four limbs in the absence of cervical pain on palpation (full range of motion not assessed). Neuroanatomic localization was to the cranial cervical spine. Beside historical tracheal collapse and an oronasal fistula, there were no significant medical comorbidities. Preliminary diagnostic tests  were unremarkable.A computerized tomographic (CT)  study of the cervical spine revealed malalignment of the atlantoaxial joint with dorsal displacement of C2 and widening of the space between C1 and C2. The dens were subjectively small in size. Additionally, the C5-6 intervertebral disc space was narrowed and showed a vacuum phenomenon. The adjacent endplates were sclerotic and mildly irregular . Based oSurgical stabilization was achieved with ventral placement of screws (self-drilling and self-tapping titanium screws)  and polymethylmethacrylate (PMMA) . A postoperative CT confirmed appropriate reduction of the atlantoaxial joint and placement of screws with PMMA . One dayThe dog presented for a recheck CT six months after surgery. The CT revealed that the cranial-most screw on the left aspect of the body of C1 was ventrally displaced about 4 mm. The atlantoaxial joint remained normally aligned, and the remainder of the screws and PMMA were unchanged. There was progressive collapse of the C5-6 disc space and ventral displacement of the cranial end plate and dorsal lamina of C6 relative to C5 . ClinicaThe dog presented to the emergency service three years later after the owners noted a new mass at the caudoventral aspect of his left ear, adjacent to the wing of C1. His neurologic examination was normal. Cervical radiographs revealed caudoventral migration of the implant and regional left-sided soft tissue swelling . The atlStaphylococcus epidermidis. The neurologic examination was normal, and the dog was transferred to the neurology service for a cervical CT scan which confirmed that the PMMA and screws had migrated ventrally and were no longer in contact with C1-C3  in addition to the clindamycin for the next 30 days. The dog presented for a recheck examination one month later and was found to be comfortable and neurologically normal. At the time of this report (17 months after surgery), the patient continues to do well.Aerobic culture of the implant showed scant growth of This case highlights the clinical, diagnostic, and surgical findings in a case of chronic atlantoaxial implant infection. The chronicity of this infection  has not been previously reported in veterinary medicine, to the best of the authors' knowledge. Long-term surgical complications described include implant migration, pin loosening and/or breakage, dyspnea, and dysphagia –5, 8. ThOne reported dog developed a ventral cervical draining tract and became nonambulatory tetraparetic 11 months after surgical stabilization with positively threaded profile pins and PMMA . This saE. coli: coagulase-positive Staphylococcus, beta-lactamase Staphylococcus and methicillin-resistant S. aureus (MRSA). One patient had no growth. It is possible that there are more described instances of implant exposure and infection with thoracolumbar implants due to less soft tissue coverage of the implant  and increased mobility of the spine at this point compared to the atlantoaxial joint. In addition, most causes of thoracolumbar injury may involve an exterior wound secondary to trauma (hit by a car or attacked by a dog) that could act as the source of infection.More chronic complications have been described with implants in the thoracolumbar spine. Commonly reported minor complications include partial screw loosening or breakage without worsening of the patient's neurologic status or evidence of implant failure , 10. MajIn humans that required revisional craniocervical junction surgeries, 13 of 55 (23.6%) patients required revisional surgery to remove an infected implant [Streptococcus bovis. This is different from the initial organism (Staphylococcus epidermidis) isolated when the draining tract was cultured. This is suspected to be due to the swab obtaining a superficial bacteria sample. In a study comparing cultures obtained from swabs and biopsies of wounds, the culture results were identical in 21.2% of the cases [S. bovis is a commensal bacterium found in the gastrointestinal tract of humans and ruminants, among other animals [S. bovis has been a notable cause of bacteremia and endocarditis. S. bovis has rarely been isolated in urinary tract infections and mitral valve endocarditis in dogs and cats [S. bovis) immediately following a dental procedure [S. bovis infection in this dog remains unknown.The culture obtained from the implant grew he cases . S. bovi animals . In humaand cats , 16. Oneand cats . In anotDespite the presence of an implant infection, the patient remained neurologically normal, with the only abnormal findings being mild cervical pain, the palpable cervical mass/abscess, and draining tract formation. This is similar to dogs in previous studies that represented mostly because of the owners' ability to palpate and/or visualize a portion of the implant under the skin or the presence of a draining tract. Besides the one patient that became nonambulatory tetraparetic, the other dogs remained neurologically normal or had mild cervical pain. This is likely due to persistent C1-C2 stabilization, and the infection primarily involving the migrated parts of the implant.In summary, atlantoaxial implant infections can occur several years postoperatively and should be considered a differential if there are physical and neurologic examination findings consistent with an abscess. Despite the presence of an implant-associated infection, patients rarely demonstrate neurologic deficits. The source of chronic infections can be challenging to determine, and obtaining a culture and sensitivity will help guide treatment with the appropriate antimicrobials. Removal of the implant is recommended to remove the nidus of infection that may be difficult for oral antimicrobials to penetrate.


Ectopic pregnancies occur when a fertilized egg implants outside the uterus, usually in the fallopian tube. Twin ectopic pregnancies are rare and pose significant diagnostic and management challenges. This case report presents the clinical details and management of a unilateral twin ectopic pregnancy in a 31-year-old female patient. The purpose of this report is to highlight the complexities associated with the diagnosis and management of this uncommon condition. In this case, we performed the left salpingectomy. We confirmed pathologically and histologically in pregnancy in the same tube. Ectopic pregnancies account for approximately 1-2% of all pregnancies and can present with various clinical manifestations. We present a case of a unilateral twin ectopic pregnancy, where both embryos are implanted in the same fallopian tube. Same time, ectopic pregnancy is one of the gynecological emergencies. It is still considered one of the most important causes of maternal death among emergency pathologies . This caA 31-year-old patient  was admitted to the clinic with acute abdominal pain and vaginal bleeding lasting four hours. The patient applied to the emergency service for the suspicion of left ectopic pregnancy in routine ultrasonography, whose gestational age was seven weeks and five days according to her last menstrual period. The patient was in good condition, conscious and well-oriented. Physical examination revealed abdominal tenderness and mild tenderness in the left adnexal region. Vaginal bleeding was mild. Ultrasonography revealed a thin endometrium and a complex mass of 32 x 42 mm adjacent to the left ovary, which is two thick-walled, fluid-filled cystic masses of 13 and 15 mm in size with further investigations.Additionally, both gestational sac containing a fetal pole with a sonographically positive heartbeat was identified within the left fallopian tube (Figure We immediately terminated the pregnancy by laparoscopic unilateral left salpingectomy under general anesthesia. Our surgical findings detected a 5 cm ectopic mass in the ampulla region. The postoperative period passed without complications. The patient was discharged uneventfully after a postoperative follow-up. Pathologically and histologically demonstrated a twin pregnancy in the same tube and Aries-Stella phenomenon detected in an endometrial cavity Figure .Although twin ectopic pregnancy is quite rare, its incidence is increasing. The literature has reported 299 cases between 1911 and 2023, and this number has been determined in the last 15 years . AssisteThe best treatment option involves various factors; hemodynamic stability, medical conditions, presence of a heartbeat, and fertility desire should be investigated. Systemic methotrexate is successfully administered to a hemodynamically stable patient with a beta-hCG value of less than 5000 mIU/ml and no fetal heartbeat . HoweverIn cases of acute ruptured ectopic pregnancy, hemodynamically unstable patients, failed medical management, or those who are contraindicated for medical management, surgical management is preferred. Laparoscopic management is becoming more popular due to its lower cost, faster recovery, shorter operation time, and hospital stay. However, salpingectomy is still the recommended treatment. In cases where fertility preservation is desired, salpingostomy may be considered. For unilateral tubal twin pregnancies, surgical management is typically the preferred treatment, as supported by the literature, similar to singleton ectopic pregnancies .The morbidity and mortality of ectopic pregnancy have decreased with improved diagnosis and treatment protocols. It has a life-threatening potential in the first three months of life. It is known to be responsible for 9%-13% of pregnancy-related deaths .Unilateral twin ectopic pregnancies are exceptionally rare, and their diagnosis and management can be challenging. Early recognition and prompt intervention are crucial in preventing complications such as tubal rupture and hemorrhage. This case report highlights the importance of maintaining a high index of suspicion in patients with an acute abdomen and the need for close monitoring and timely surgical intervention. Further research and clinical reports are warranted to improve our understanding of this uncommon condition and optimize its management.


An abnormal mass in the head and neck involving the supraglottic and cervical region offers a wide range of differential diagnoses. The pathology is either benign or malignant in nature. Castleman disease (CD) is an uncommon lymphoproliferative disorder characterised by hypervascular lymphoid hyperplasia and is classified into unicentric or multicentric disease. Histopathologically it is divided into hyaline vascular (HV), plasma cell (PC), and mixed cellularity variants. The multicentric disease is linked with PC and has the propensity to progress to lymphoma or Kaposi Sarcoma. We report a case of a 45-year-old gentleman who presented with a painless anterior neck swelling and left supraglottic mass for six months. Computed tomography (CT) contrast imaging demonstrated a homogenous enhancing lesion at the left supraglottic and the midline of the anterior neck with erosive changes of the thyroid cartilage. A surgical resection of the anterior neck mass was performed. The diagnosis of Castleman disease plasma cell variant was made by histopathologic evaluation. The patient remained well post-resection. Supraglottic multicentric Castleman disease is the least expected diagnosis in this case. Unicentric disease is treated with surgery. However, limited studies are available in determining the effectiveness of surgery in multicentric diseases. The plasma cell variant requires a multidisciplinary and multimodal approach due to an inclination towards malignancy. Research is needed to determine the role of surgery in multicentric disease and to develop optimum guidelines for managing cases. To date, there is unsubstantial literature describing supraglottic multicentric disease. It has been acknowledged that this lymphoproliferative disorder is caused by human herpes virus infection of the B-cell pool and the lymphovascular compartment of lymph nodes (human immunodeficiency virus (HIV) infection, with an estimated 50% occurring in seronegative HIV cases (Castleman disease (CD) is a rare benign lymphoproliferative disorder, primarily characterised by progressive lymph node enlargement with an estimated incidence of 15.9-19.1 cases per million person-years (ph nodes -5. Much IV cases .Human herpes virus-8 (HHV-8) may play a role in the pathogenesis of MCD  revealed otherwise. CD of the head and neck can be confused with other neoplasms due to its paucity of signs and symptoms. This case report discusses the variation of clinical presentation and the recommended management of CD of the head and neck in HIV-negative MCD. Our literature review shows a few published articles describing HIV-negative plasma cell (PC) type MCD involving the head and neck region. However, no reports are available reporting PC type MCD specifically related to the supraglottis.A 45-year-old Chinese male presented with six months history of an anterior neck swelling that gradually enlarged over two months. It was painless and not associated with any disturbance in deglutition, breathing or speech production. There were no other related symptoms that were suggestive of infective aetiology. Physical examination revealed a 3cm-by-3cm anterior midline neck mass at the level of the thyroid gland . The mass moved with tongue protrusion and swallowing. A flexible fibreoptic nasolaryngoscopy revealed an obliterated left pyriform fossa. Fine needle aspiration cytology (FNAC) of the midline mass reported atypical mononucleated cells suggestive of neoplastic lesion. An ultrasound (US) guided tru cut biopsy was carried out to get further tissue for confirmation. Formal results reported back as reactive plasmacytosis. The contrast-enhanced computed tomography (CT) scan demonstrated a homogenously enhancing lesion at the left supraglottic region measuring 2.3cm x 1.7cm x 2.5cm, extending from the level of the hyoid bone to the level of the false cord inferiorly and posteriorly to the anterior commissure, causing widening of the paraglottic fat with medialisation of the mucosal space .The plane between the left strap muscles with this mass at hyoid and infrahyoid bone level is lost, displaying erosive changes involving the left thyroid cartilage . The inferior aspect of the left aryepiglottic fold is thickened, obliterating the left piriform sinus. Another well-defined homogenously enhancing lesion at the anterior midline neck at thyroid cartilage level measures 1.3cm x 2.5cm x 2.1cm. Subcentimeter cervical lymph nodes bilaterally were present at levels Ia, Ib, II and III. There was no prominent lymphadenopathy seen in the mediastinum. He underwent a direct laryngoscopy (DL), anterior neck dissection and selective neck dissection. DL revealed an oedematous left epiglottis and arytenoids, and a biopsy was taken . Intraoperatively, there was a mass at the anterior neck 4cm x 3cm below the skin (with skin tethering) and anterior to strap muscles. The anterior neck mass, fibrofatty tissue, and bilateral level 1b was removed en bloc .Histopathology examination (HPE) of the supraglottic mass reported infiltration of deep subcutaneous tissue with dense lymphoid infiltrates composed of varying sizes of lymphoid follicles with hyperplastic germinal centres. The interfollicular area was expanded with numerous reactive plasma cells .The Ki67 proliferative index was ~5%. The lymphoid cells are positive for cluster of differentiate 20 (CD20). The germinal centres are highlighted by CD 10. The follicular dendritic cell (FDC) meshwork is highlighted by CD21. He has recovered well after surgery and remains in remission on further follow-up. In addressing the remaining supraglottic mass, he was counselled for partial pharyngectomy for therapeutic purposes. However, the patient’s immense concern about the potentially associated surgical morbidities has hindered him from pursuing further completion of the resection of the supraglottic mass. He remains well today.The clinical characteristic of this case demonstrates that reaching a final diagnosis has proven to be a challenge to the surgeon, despite obtaining FNAC and tru cut biopsy results. This is due to the non-specificity of the presenting symptoms with a midline neck mass that could lead to a misdiagnosis, such as a thyroglossal duct cyst or a thyroid nodule/cyst. The signs and symptoms in CD are atypical and rarely present in the head and neck region . CD has Two main histopathology variants have been identified: hyaline vascular (HV) type 75-90%), plasma cell (PC) type 10-25%) and thirdly, variable degrees of mixed cellularity features in-between (15%) . These features are specific in patients with the PC type MCD ,16. The disease presentation in HIV-negative patients can be categorised as either HV type UCD or PC type MCD. In a systemic analysis by Talat et al. and Taiqin et al., the authors identified characteristics associated with PC type MCD. The age distribution presentation of PC type MCD is between the fifth and sixth decade of life, with higher occurrences in male patients ,12. For Hepatosplenomegaly is present in most PC type MCD patients . AbnormaA concise clinical workup is crucial in determining classification and staging. Pre-operative investigations can be challenging because FNAC and radiology studies are of limited value in concluding the diagnosis. CD can mimic other lymphoproliferative diseases and neoplastic disorders even when the differential diagnosis list is narrowed down. The patient should undergo a clinical examination of the peripheral lymph node that is most likely to be involved. Remotely located or visceral lymph nodes should also be investigated to exclude from CD. The best method would be a whole-body contrast CT scan directed at the neck, chest, abdomen, and pelvic regions . The priSurgery is the gold-standard treatment of UCD with or without radiation therapy and has an overall survival rate of 95%. . The priManagement of MCD is utterly different from UCD. The clinician is faced with an entirely different setting. The clinician's role is limited; first, it is to gain tissue for diagnostic purposes, and second, to debulk the dominant foci of multicentric disease when there is the presence of life-threatening complications such as vascular or airway compromise, massive organomegaly, or bowel obstruction . Bowne e16 Rituximab is known to exacerbate Kaposi Sarcoma (Hence, a multimodal therapy is recommended, which includes debulking surgery or resection when indicated, combined with single or multi-agent chemotherapy, followed by antiviral therapy for disease control and monoclonal antibody therapies targeting CD20 or IL-6 ,15. Ritu Sarcoma . Cytotox Sarcoma . There i Sarcoma , but the Sarcoma ,20,21. I Sarcoma . While O Sarcoma . A 32% o Sarcoma ,17. In a retrospective study involving five HIV-positive PC type MCD, two patients were simultaneously diagnosed with Kaposi sarcoma, and another eventually developed Kaposi sarcoma .CD seldom involves the supraglottic and neck region. A supraglottic mass and cervical lymphadenopathy warrant a vast list of differentials, including malignancy and lymphoproliferative diseases, and the least expected differential diagnosis, in this case, is MCD involving the supraglottis. Most cases are HV type UCD, with a significant incidence in HIV-positive cases. UCD and MCD are two different conditions with diverse patient characteristics, manifestations, variable responses to therapy and long-term outcome. Acknowledgement of the subtypes of CD determines the surgeons’ role and approach in the diagnosis and treatment strategy. Surgery is the gold standard for treating UCD, but in MCD, it is a whole different picture. The patient's refusal to resect the supraglottic mass leaves the surgeon in a dilemma. The main objective is to provide the best outcome for the patient. PC type MCD requires close observation with a multidisciplinary and multimodal approach to treatment, which might be managed with chemotherapy, as there is an inclination towards malignancy. Further research is needed to determine the aetiology and pathogenesis of CD, to enable the development of enhanced and novel therapeutic options for this rare disease.Author Contributorship StatementAFJ interviewed, reviewed clinical notes and data, and wrote and submitted the manuscript. MRMY initiated the case report, provided images, and proofread and edited the manuscript. DJY contributed to data collection, providing patient images and editing the manuscript. MMB proofread and edited the manuscript. RRMZ provided histology slides, given comments on the histopathology section and edited the manuscript. All authors have read and approved the final manuscript.