Adrenocortical carcinomas are extremely rare in the paediatric population. Most of them are hormone-secretive lesions; therefore, they should be taken into consideration in a child with signs of precocious puberty and/or Cushing’s syndrome symptoms. Nonetheless, differentiation from benign adrenal tumours is necessary. We report a rare case of adrenocortical carcinoma in a girl and a literature review using the PubMed database. A four-year-old girl presented with rapidly progressing precocious puberty and signs of Cushing’s syndrome. Imaging of the abdomen revealed a large heterogeneous solid mass. Histopathologic evaluation confirmed adrenocortical carcinoma with high mitotic activity, atypical mitoses, pleomorphism, necrosis, and vascular invasion. After tumourectomy, a decrease of previously elevated hormonal blood parameters was observed. Genetic tests confirmed Li Fraumeni syndrome. Adrenocortical carcinoma should be suspected in children with premature pubarche and signs of Cushing’s syndrome. Diagnosis must be based on clinical presentation, hormonal tests, imaging, and histopathological evaluation. Complete surgical resection of the tumour is the gold standard. Oncological treatment in children is not yet well-studied and should be individually considered, especially in advanced, inoperable carcinomas with metastases. Genetic investigations are useful for determining the prognosis in patients and their siblings. Adrenocortical tumours (ACT) including adrenocortical carcinomas (ACC) and adrenocortical adenomas (ACA) are rare in the paediatric population, with an incidence of 0.3–0.38:1,000,000, accounting for approximately 0.2% of all paediatric neoplasms .ACT are more common in women, with a proportion of 1.4:1. This rate varies according to age, but remains higher in women , 4, 5, eA much higher percentage of ACT is observed in Brazil, with an incidence of 6.2:1,000,000 in children <10 years , and forThe majority of ACT and ACC are sporadic; however, there is an increased risk of developing the tumour in patients with genetic syndromes such as Li Fraumeni syndrome, Beckwith–Wiedemann syndrome, multiple endocrine neoplasia type 1 (MEN-1), familial adenomatous polyposis, and other hereditary cancers , 9, 10.Most ACT in the paediatric population are hormone-secreting lesions , 2. The Non-functional ACT and ACC are much less common. The manifestation of adrenal tumours is non-specific, including abdominal or back pain, fatigue , 5, or aThe differentiation between benign adrenocortical adenomas and ACC presents a challenge at every stage of the diagnostic process, including clinical symptom analysis, laboratory tests, imaging scans, and histopathologic evaluation , 13, 14.The only undisputed sign of malignancy is metastases. The most frequent sites of ACC metastases are the lung, liver, and retroperitoneal space . MetastaThe aim of this study was the presentation of a rare cancer patient and a review of the up-to-date knowledge of adrenocortical carcinoma and its diagnostics-therapeutics algorithm.A four-year-old female presented with rapidly progressive precocious puberty. Pubic hair was first observed approximately two months prior to presentation. Additionally, the child’s mother noticed intense perspiration and acne. The patient gained approximately 3 kg in one month and became apathetic.The patient’s medical history was insignificant: second pregnancy, second delivery , Apgar score of 10, birth weight of 3,900 g, and birth length of 55 cm. Psychomotor development was normal. She had no chronic diseases, no allergies, or no operations in the past. Her diet was normal and physical activity was regular; however, she became less active in the weeks prior to presentation.On physical examination, the patient’s general condition was good, her vital signs were stable, her height was 110 cm (90th percentile), her body weight was 23.9 kg (>97th percentile), and she appeared overweight with an excess of visceral adipose tissue, a rounded face, and prominent, reddened cheeks. She also had acne Figure 1The abdomen was asymmetrically distended and a firm, non-tender, and immovable mass was palpated on the left side. Physical examination also revealed pubarche, Th1 P3 A1 on the Tanner scale.Abdominal ultrasonography (USG) revealed a large heterogeneous solid mass between the spleen and left kidney, measuring 121 x 100 x 141 mm, with several blood flow signals. The mass shifted and modelled surrounding organs. Additionally, a small round lesion of unidentified character was observed in the right lobe of the liver.Computed tomography of the abdomen showed a large left suprarenal mass measuring left-right (LR) 10 cm; anterior-posterior (AP) 13.5 cm; and cranio-caudal (CC) 12 cm, with calcifications 3. The tThe patient’s bone age was six years based on the Greulich-Pyle method.Laboratory investigations are shown in The girl was qualified for surgical intervention. Before surgery, she was prepared with hydrocortisone in high substitutive doses. The patient was referred to the Department of Surgery, The Children’s Memorial Health Institute, where she underwent radical tumourectomy, with resection of the left kidney and the lesion in the liver. After the tumourectomy, the high doses of hydrocortisone were continued. The perioperative period was complicated by lymphorrhoea, pancreatitis, pneumonia, left side pleural effusion, and gastrointestinal obstruction that required a second laparotomy. In the subsequent days, the patient improved. The substitution of hydrocortisone was decreased, and fludrocortisone was introduced orally.Histopathologic evaluation of the tumour confirmed ACC with high mitotic activity, atypical mitoses, pleomorphism, necrosis, and vascular invasion. The structure of the left kidney was normal. No metastases were found in the local lymph nodes. The lesion in the sixth liver segment was confirmed to be a cavernous angioma.In the post-operative follow-up, the patient remained under observation of the Department of Oncology in The Children’s Memorial Health Institute, Warsaw. In a control CT of the abdomen, chest, and pelvis, no lesions were observed. The control steroid profile showed normal daily excretion of androgens and 17-OH-progesterone metabolites.At the request of her parents, the patient was transferred to the Department of Haematology, Oncology and Transplantology of the Children’s Hospital of Lublin to continue treatment. At this stage, no new symptoms presented. The development of the girl is normal: her Tanner scale is Th1P2A1. The second control steroid profile was normal.Genetic investigations confirmed Li Fraumeni syndrome in the girl. There is a need to examine the patient’s siblings. The parents did not consent to oncological treatment of their daughter.ACT, encompassing both benign and malignant lesions, are rare in the paediatric population, accounting for 0.2% of paediatric neoplasms. The literature reports the peak incidence of ACT in children at either <five years, or at a biphasic age distribution, with the first peak <five years and the second >10 years .At the molecular level, two common aberrations are described—excessive expression of IGF-2 and constitutive activation of the signal route Wnt/β-catenin , 17, 18.It was recently observed that germline EGFR variants  and vitaClinical manifestation of ACT is different in children and adults. Adults present with non-specific symptoms more often than hormonal hypersecretion due to the tumour-mass effect , while cThe most frequent symptom of ACC in children is virilisation, which is characterised by precocious development of pubic or axillary hair, penis enlargement or erection, clitoris enlargement, accelerated growth velocity with high bone-age, acne, hirsutism, and voice changes. The second most common manifestation is Cushing’s syndrome, with symptoms such as central obesity, moon face, and hypertension , 22, 23.The appearance of precocious puberty should always prompt consideration of pathology within the adrenal cortex in the diagnostic process , 24–26. Laboratory diagnostics of ACC are based on hormonal activity observed in over 90% cases. Clinical manifestation results from excessive secretion of steroids and their precursors by the tumour. The most common abnormalities are increased levels of DHEAS, androstenedione, testosterone, 17-OH progesterone, and 11-deoxycorticosterone , 24. AmoUltrasonography (USG) of the abdomen is the first stage of imaging in a patient with clinical signs of adrenocortical pathology. Computed tomography (CT) is the next step in the diagnostic process , 2 and iAccording to Kuhlen et al. , howeverMalignant tumours are usually bigger than benign ones; however, diagnosis cannot be based only on dimensions. Nonetheless, tumours ≤5 cm seem to be benign, while tumours ≥10 cm are more likely to be malignant, especially with a concomitant elevated LDH level .On imaging, ACA have homogenous contrast enhancement and are well circumscribed, while ACC are usually characterised by a heterogenous structure with necrosis, haemorrhage, and calcifications. ACC can also invade local structures and vessels . The preTumour biopsy is not recommended because resection is a basis of treatment, and tumour rupture and spillage during biopsy worsen the prognosis .There is no single histopathologic trait that can reveal the malignant character of adrenal tumour, apart from invasion of nearby tissues. Nonetheless, architectural disarray, reticulin framework disruption pleomorphic, and large, clear, and granular or eosinophilic neoplasm with intranuclear inclusion and nuclear atypia may help differentiate between benign and malignant adrenal tumours and are indications of ACC rather than ACA .Children with ACT have better prognosis than adults ; howeverTumour weight >400gTumour size >10.5cmExtension into periadrenal soft tissues and/or adjacent organsInvasion into vena cavaVenous invasionCapsular invasionPresence of tumour necrosis>15 mitoses per 20 high power field (400X)Presence of atypical mitotic figuresAccording to Wieneke’s criteria, it is possible to determine the prognosis outcome of the patient. Two criteria indicate a benign long-term clinical outcome, three criteria indicate an intermediate/atypical/uncertain malignant potential while four or more criteria indicate a poor clinical outcome. , 36.It was observed that the cellular proliferation index Ki-67 was significantly different between ACC and ACA, with a mean level of 30.2% (range 7–80%) and 9.9% (range 2–20%), respectively . In anotFang et al. suppose that ACC development may depend on intracellular communications mediated by miRNA and mRNA .According to the European Network for the Study of Adrenal Tumours (ENSAT), there are four stages of ACC. Stage I and stage II are strictly localised tumours with a size of ≤5 or >5 cm, respectively. Stage III is characterised by infiltration of surrounding tissue, positive regional lymph nodes, or a tumour thrombus in the vena cava and/or renal vein. Stage IV is defined by the presence of distant metastasis , 4. StagComplete surgical resection is the mainstay in ACT treatment as it enables full control of the disease and satisfactory clinical effects, especially in cases that are not advanced , 42–44. In malignant adrenal tumours, open surgery rather than laparoscopic should be prioritized . IncreasThe effect of adjuvant chemotherapy in treating ACC is not satisfactory; adrenal cancer cells are resistant to common drugs. Mitotane, an inhibitor of the adrenal cortex, is commonly used in adult patients with ACT , 42, 44.Toxicities related to combined therapy with mitotane and chemotherapy may lead to discontinuation of treatment , 48, 49.During mitotane treatment, substitutive doses of hydrocortisone should be 2–3 times higher than those used for congenital, adrenal hyperplasia (CAH), and in some patients, fludrocortisone treatment for control of electrolytes is necessary. Serum mitotane evaluation should also be reported as well as the dose, usually every 15 days , 44, 47.ACC seems to be sensitive to radiotherapy (RT). Adult patients who received RT as an adjuvant treatment had higher overall survival (p=0.004) than patients treated with surgery alone . WiegeriCurrently, the role of immunotherapy in ACC treatment in adult patients is being investigated. The potential use of immune checkpoint inhibitors, such as pembrolizumab, which has a favourable safety profile and good tolerance in initial evaluation in adult patients , 52, 53,ACC should be suspected in children with premature pubarche and signs of Cushing’s syndrome. For diagnosis, hormonal tests and imaging  are necessary. The surgical treatment of ACC is the gold standard, and oncological treatment should be individually considered as the second adrenal gland may be destroyed. Genetic investigations are useful for determining the prognosis in patients and siblings.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving humans were approved by Ethical Committee of Medical University in Lublin. The studies were conducted in accordance with the local legislation and institutional requirements. The human samples used in this study were acquired from a by-product of routine care or industry. Written informed consent for participation was not required from the participants or the participants’ legal guardians/next of kin in accordance with the national legislation and institutional requirements. Written informed consent was obtained from the minor(s)’ legal guardian/next of kin for the publication of any potentially identifiable images or data included in this article. Written informed consent was obtained from the parents of the child for the publication of this case report.EZ, MM, AG, and IB-S made substantial contributions to the conception, design, and acquisition of data, drafting of the article, giving final approval of the version to be published, and agreeing to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. IB-S critically revised the paper for important intellectual content. All authors contributed to the article and approved the submitted version.


Sclerosing meningiomas (SMs) represent a rare histological variant of meningiomas, first described in 1989 as invasive bulking masses of whorling collagen bundles with a minimum percentage of meningothelia-resembling cells, and they are often misdiagnosed. The literature reports only 30 cases of SMs, with only two of them being intramedullary. The authors present the case of a patient with a cervical intramedullary SM who presented with gait disturbances, sensory deficits, weakness in four extremities, and hyperreflexia. The surgery was performed under neurophysiological monitoring and after administration of sodium fluorescein, which allowed us to discriminate the exact myelotomy point. Intramedullary SMs are very rare entities whose correct management may result in a good outcome. We present here a case of a 66-year-old woman who was admitted to our institution with a 6-month history of progressive extremities weakness, paresthesias of both superior arms, worse on the right side, followed by gait and balance disorders. She has a history of meningiomatosis where a huge convexity meningioma was operated 5 years early without any neurological impairment. At the neurological examination, the patient exhibited a decreased general muscle strength with a 3/5 MRC scale quadriparesis, a bilateral C4–5 territory hypesthesia. Babinski reflex was positive with a generalized hyperreflexia of deep tendon reflexes. Romberg test was positive.2 In case of high risk of surgical morbidity, less invasive treatments are justified and they are tailored on the specific lesion in order to achieve the best surgical outcome and the best prognosis for the patient. Modern functional neurosurgical tools as intraoperative neurophysiological monitoring could give the surgeon the opportunity to reduce neurological deficit guaranteeing the gross-total resection. The use of fluoroscopes during the procedure could improve the curative goal of surgical approach. Benefits of the surgical procedure are biopsy and diagnosis, gross-total resection with no need of other treatments, neurological function recovery, arrest of further progression. Risks are vascular injuries, neurological impairment to achieve radical surgery, CSF leak, wound complications. Alternatives are the neuroradiological follow-up during time. The surgery was made with a patient in a prone position with a preoperative administration of fluorescein. Preoperative x-ray could help us to localize the lesion level. The surgery was made with the intraoperative neurophysiological monitoring. The patient underwent a posterior bilateral C4–5 hemilaminectomy and microscopic exeresis of the cervical lesion.The total spine MRI showed an intradural intramedullary lesion at C4–5 level. The lesion was hyperintense in T1- and hypointense in T2-weighted sequences. After administration of gadolinium, the lesion demonstrated heterogeneous contrast enhancement with a hypointense core. The benign nature of the vast majority of meningiomas defines the surgical strategies as the gold standard treatment of these lesions. Gross-total resection with a one-step surgery is curative.The exeresis was performed under neurophysiological intraoperative D-wave, MEPs and SEPs monitoring, and after administration of sodium fluorescein, which allows us to intraoperatively discriminate the intra-axial nature of the lesion. Tumor was found to be white-grayish, pretty avascular, with a hard consistency and without an arachnoidal plane among the medullary parenchyma, especially in the ventral plane. The intraoperative pathological examination documented a paucicellular sclerotic bulk mass with a moderate vascular component.4 Intramedullary sclerosing meningiomas are not mentioned in the WHO 2021 CNS tumor classification, and they are included into other morphological variation subtype group. As far as WHO 2016 added the criterion of brain invasion to atypical meningioma diagnosis, we must reconsider their clinical significance.3The microscopic analysis demonstrated a hypocellular proliferation with a sclerotic stroma, a scanty population intermingled by cellular areas with spindle elements, arranged in intersecting fascicles and a variable collagen stroma. The immunohistochemical evaluation demonstrated a strong stain for vimentin, focal positivity for epithelial membrane antigen, positive nuclear staining for progesterone receptors, and a very low proliferation demonstrated by Ki-67 < 2%.5 The 6-month postoperative neurological examination showed an improved four-extremities strength with no alteration in the segmental proofs. Sensory alterations were improved with no gait and balance disorders complaint. Romberg test was negative. Paresthesias were not described by the patient.The postoperative MRI demonstrated the residual surgical cave after the administration of gadolinium.The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this publication.Primary surgeon: Cardali. Assistant surgeon: Garufi, Ricciardo. Editing and drafting the video and abstract: Garufi, Ricciardo, Conti. Critically revising the work: Garufi, Conti. Reviewed submitted version of the work: Garufi. Approved the final version of the work on behalf of all authors: Garufi. Supervision: Garufi.The necessary patient informed consent was obtained in this study.


One-third of all intestinal obstructions in the newborn are caused by atresias. The most common site is the duodenum followed by jejunoileal and colonic locations. Herein we report the autopsy findings of a rare case of jejunoileal atresia associated with malrotation of gut. Autopsy performed on a 36 weeks old male fetus still birth, born of a non-consanguineous marriage, demonstrated jejunoileal atresia with apple peel deformity and malrotation of gut. Although the diagnosis was established in the prenatal period, in utero fetal demise occurred before definitive surgical intervention could be done. This case highlights the importance of early diagnosis and intervention. Intestinal atresia is a rare congenital anomaly, the incidence of which ranges from 1.3 to 3.5 per 10,000 live births . Twenty A 2.22 kg male stillborn neonate was delivered vaginally following induction of labour to 31-year-old gravid 3, para 2 mother at 35 weeks 5 days period of gestation. The parents were in a non-consanguineous wedlock. The mother was a known case of hypothyroidism on eltroxin. There was previous history of still birth during first pregnancy. Dual and triple screen tests were negative. Maternal antenatal routine investigations were essentially normal. Antenatal ultrasonography in the first and second trimesters was normal. However, during a routine visit in the third trimester, the ultrasound scan showed a dilated gastric fundus bubble, and dilated proximal bowel with a maximum diameter of 2.5 cm A. No assPertinent findings on autopsy included a macerated male fetus whose anthropometrical measurements were normal for age and sex. Development of facial features, thorax, and extremities appeared appropriate for the gestational age. No anatomic defects were identified on gross evaluation. Head, chest and abdominal circumference were normal for 35 weeks gestational age. Cephalhaematoma with overriding of skull bones was noted. On opening the body, the most remarkable finding was marked distension, dilatation and congestion of the duodenum and jejunum that ended in a blind pouch B,C. The Histopathological examination of most of the organs showed autolytic changes Sections from the dilated duodenum showed autolytic mucosa with a markedly thickened muscle layer C. SectioFetal tissue submitted for cytogenetic analysis showed normal cytogenetics.It is certified that ethical approval of ethics committee of Command Hospital Air Force Bangalore and consent of next of kin of deceased was sought prior to publication of this study.One-third of all intestinal obstructions in the newborn are caused by atresias ,2. The aJejunoileal atresia could be single >90%) or multiple and may be familial 0% or mul. Our casThe most commonly accepted theory for pathogenesis of intestinal atresia is based upon work by Louw and Barnard ,14. TheyWhat might have caused intrauterine fetal demise in the current case? It is postulated following obstruction, the raised bile acids in fetal bloodstream, may have triggered a fetal arrhythmia, or marked dilatation of the stomach, duodenum and proximal jejunum in this case may have triggered bradycardia and ultimately sudden death resulting from high vagal tone ,16.With respect to prenatal diagnosis, repeated ultrasound imaging is useful to assess the progress and development of atresia in the foetus. If undetected, the neonate may present with signs and symptoms of high bowel obstruction several hours after delivery. Despite the high mortality, it can be treated and managed with appropriate early surgical intervention .Our autopsy case report presents a rare and complicated variant of jejunoileal atresia associated with malrotation of gut. This case highlights the importance of early diagnosis and intervention.The authors declare no conflict of interest.None


Persistent hyperplastic primary vitreous (PHPV), also known as persistent fetal vasculature (PFV), is a clinical entity that traditionally presents with leukocoria, microphthalmia, retinal dysplasia, or eyeball shrinkage which is associated with poor vision. However, there is a dearth of literature on cases of PHPV in adulthood or with asymptomatic occurrence. This report presents the clinical and pathological findings of a non-typical PHPV case and discuss the current knowledge for this condition.A 68-year-old healthy male was referred to our outpatient department for evaluation of age-related cataract without other visual symptoms. Preoperative fundus examination occasionally detected an isolated stalk-like band extending to the posterior pole of the eye with normal central vitreous and retina. Other ocular examinations including b-mode ultrasonography, optical coherence tomography did not unveil any abnormalities, which caused diagnostic uncertainty. We referred to cataract surgery along with histopathological study, that revealed characteristics of PHPV including fibrous connective tissues mainly composed of fibrocyte proliferation and a very few capillary vessels. Thereafter, a definitive diagnosis of non-typical PHPV was established.Our case is unique due to it was not discovered until adulthood, presence with only age-related cataract, and accompanied with normal central vitreous and retina. Histopathological explorations lead to an accurate diagnosis of the condition. Those results broaden the phenotype spectrums of PHPV and further provide clinical clues for the cognition of the disease. Persistent hyperplastic primary vitreous (PHPV), also known as persistent fetal vasculature (PFV), is a rare congenital malformation of the eye resulting from continuous proliferation of the original vitreous and hyaloid vasculature, which failed to regress normally during the embryonic period , 2. It iThe knowledges of PHPV have evolved over time. Recent studies reported the involvement of various signaling pathways in the pathogenesis of PHPV, including protooncogene ski, tumor suppressor gene Arf, p53, Bax and Bak, ephrin-B2, ephrin-A5, FZD4 and LRP5 –10. BesiThe heterogeneity of this disease makes diagnosis challenging. The definitive diagnoses of PHPV are typically established according to b-mode ultrasonography, color doppler ultrasound, and radiological investigations including CT and MRI imaging , 13, 14.In this study, we describe the case of a 68-year-old otherwise healthy male patient who complained of merely age-related cataract to our outpatient department without other visual symptoms. During preoperative routine fundus examination, a gray, stalk-like band extending peripherally into the vitreous cavity was occasionally detected. However, the results of B-scan ultrasound and other clinical examinations failed to yield a definite diagnosis. Thus, we referred to histopathological explorations of the patient’s tissue obtained during cataract surgery through which an ultimate diagnosis of atypical PHPV was established.Herein, we will present the clinical and pathological findings of this unusual case and discuss the diagnosis and therapeutic options available for this condition. This case was unique due to it was not discovered until adulthood, as well as mild clinical characteristics. The results broaden the phenotype spectrums of PHPV and further provide clinical clues for the cognition of the disease.A 68-year-old male presented to our outpatient department in April 2022 with a chief complaint of blurred vision in his left eye for 1 year. He denied a history of infection, trauma, and ocular surgery. There was no antenatal, postnatal, or familial history related to the disease. During the ophthalmological examination, his left eye was found to have age-related cataract with a best corrected visual acuity (BCVA) of 20/100. The cornea was clear, the anterior chamber was normal, the pupil was round and regular, and it reacted normally to light  based on the corneal tomography performed with Scheimpflug imaging . Consequently, the patient was subjected to a toric intraocular lens (IOL) implantation in his left eye. Preoperative marking of the toric IOL axis was conducted with the patient in an upright position to avoid misalignment due to cyclotorsion using the Robo Marker .Intraoperatively, after removal of the lens, a floating fibro-vascular stalk-like band attached to the very peripheral, mainly at the nasal part of the posterior lens capsule and extended its free tail into the vitreous cavity was detected. The posterior capsule ruptured when the viscoelastic substance was injected into the capsular bag  staining. Histopathological examinations demonstrated that the tissue was a fibrous connective tissue mainly composed of proliferation of fibrocytes, and a very few capillary vessels Fig. B. These Postoperatively, the patient was treated with a standard protocol consisting of topical antibiotic and anti-inflammatory therapy agent for a duration of 2 weeks. We followed up with the patient for 5 months. The BCVA in his operated eye improved and remained in 20/20, with a transparent cornea, normal anterior chamber, a round pupil, normal IOP, and with the IOL in the proper location Fig. A, a normPersistent hyperplastic primary vitreous (PHPV) is a rare congenital ocular abnormality caused by the continuous proliferation of the original vitreous in the embryonic stage without degeneration . The cliOne of the unique features of our patient is that the fibrovascular stalk persists into adulthood until it is discovered. In the fetus, the original vitreous of the eye is formed in the seventh week of gestation and contains branches of the hyaloid artery between the crystalline lens and retina. During later stage, the original vitreous and hyaloid vasculature are replaced by avascular secondary vitreous, which initiates in the 20th week of gestation and completes at birth . As has Another unusual feature is that our case constituted only an isolated stalk-like fibrovascular band extending to the posterior pole combined with normal central vitreous and retina. Based previous report, the vast majority of PHPV cases have a combination of anterior and posterior segment involvement . AnterioThirdly, one more rare feature of the current case is that the patient is asymptomatic and denied any vision impairment in early childhood. Predictors of poor visual prognosis in PHPV include bilaterality, microphthalmos, glaucoma, and posterior segment involvement, which will lead to an increased risk of retinal detachment or even eyeball shrinkage. Posterior PHPV usually leads to a poor vision prognosis, according to previous studies , 5. The Clinically, the diagnosis of PHPV is usually made based on typical clinical signs and imaging manifestations. It is now accepted that the b-scan ultrasonography, color doppler ultrasound, CT, and MRI are vital for disease diagnosis, especially in eyes in which the posterior segment is not visible , 28. TheSurgery was indicated for the patient due to the presence of cataract. During surgery, the fibrovascular stalk was observed to be connected to the posterior lens capsule, which ruptured due to the dragging of the fibrous membrane. This is a known complication of PHPV and is often ascribed to the invasion of the abnormal fibrovascular tissue which exerted increased traction on the posterior capsule . As a reIn conclusion, we reported a non-typical case of PHPV in a 68-year-old adult, he was previously healthy, presented with only age-related cataract, and accompanied with normal central vitreous and retina. Our case broadens the phenotype spectrums of PHPV and provides clinical clues for the cognition of the disease. More importantly, we successfully diagnosed this non-typical PHPV case with a histological approach. We suggest that a similar approach may be considered for this patient group.


Median rhomboid glossitis (MRG), also known as posterior midline atrophic candidiasis, is a developmental disorder of the oral cavity. It usually involves the central part of the dorsum surface of the tongue. It is a papillary atrophy that is generally well-defined and symmetrical in presentation. It is usually a painless lesion, but some patients may complain of mild itching and irritation in that region. This lesion is more predominant in males than females. It has been widely argued that this lesion is not a developmental disorder but a clinical manifestation of a fungal etiology. This article presents a case report of a 55-year-old woman who reported to the oral medicine department in a tertiary care center in Wardha, India, with the chief complaint of an oval-shaped, discolored area on the dorsum of her tongue since birth. This article also emphasizes the role of a dental practitioner in diagnosing the lesion and appropriate patient education regarding the condition. There are many developmental disorders of the tongue that result in its depapillation. Hence, it is very important that the practitioner who is diagnosing the condition has adequate knowledge about all the diseases and conditions that are related to the oral cavity ,2. MediaA 55-year-old woman reported to the oral medicine outpatient department with the chief complaint of a patch of discoloration on the dorsum of her tongue since birth. The patient noticed an oval-shaped patch of pink at the center of her tongue when she was about 12 years old. She had previously visited three dentists and one dermatologist in the surrounding area, but they could not diagnose the condition. She was then referred to the current hospital. The lesion was asymptomatic, and no discomfort or irritation was reported. On intra-oral examination, a depapillated area was seen in the center of the dorsum of the tongue anterior to the circumvallate papilla of about 1 cm × 2 cm in size, roughly oval in shape; the surface was smooth; and the margins were well demarcated. On palpation, there was no local rise in temperature or tenderness present. Figure On the basis of the history given by the patient and the clinical examination performed by the clinician, MRG was diagnosed. Based on the oral surgeon’s perspective and considering all the factors, the patient was advised that no invasive investigation like a biopsy should be done as the lesion is painless, long-standing, and not suspected of carcinoma. The patient was educated about the nature of the lesion, and no treatment was advised as the patient was asymptomatic.Median rhomboid glossitis is a rare developmental condition of the tongue impacting 0.01-01% of the population . It is mMedian rhomboid glossitis (MRG) is an often unnoticed and asymptomatic developmental disorder of the tongue. This lesion often goes undiagnosed up until the middle ages of the person; decent knowledge regarding diseases of the tongue is needed to effectively come to a diagnosis; hence, the role of healthcare professionals like dentists is important as they are the ones who primarily deal with the oral cavity and can diagnose the disease during the very first oral examination of the patient. If there is a dilemma regarding the nature of the lesion, a biopsy should be advised. If lesions are symptomatic, then antifungal therapy is advised, but in cases where the lesion is asymptomatic, no treatment is necessary.


Meningiomas are extra-axial neoplasms that originate from the arachnoid cap cells located on the inner surface of the meninges. Approximately 36% of central nervous system tumors are meningiomas. Based on earlier findings to be benign in most cases, they are categorized as slow-growing tumors that form gradually over time. Meningiomas are usually asymptomatic and discovered inadvertently. They rarely present with immediate clinical symptoms or abrupt hemorrhagic strokes. However, tumor hemorrhage can be fatal in high-grade meningiomas, particularly those with vascularization. We describe a 58-year-old man who was hospitalized after experiencing an unexpectedly acute headache. The right cerebellar hemisphere and vermis cerebellar hemorrhage were detected on computed tomography (CT), and the cerebellar hemorrhage was explained by a diagnosis of hypertension. When additional analysis of the patient’s chest CT indicated lung mass lesions, we assumed that the lung cancer had spread to the brain. However, the pathological outcomes of a guided definite pulmonary aspiration biopsy, in conjunction with resection of the cerebellar tumor, suggested a subtentorial meningioma with ruptured hemorrhage and pulmonary meningioma metastasis. The patient was transferred to a hospital closer to home for ongoing follow-up and, after 2 months, he had recovered well. TheHerein, we present the case of a middle-aged man who had a sudden headache and was initially diagnosed with hypertensive cerebellar hemorrhage on head computed tomography (CT). However, following additional magnetic resonance imaging (MRI) of the head, and chest CT, the diagnosis changed to lung cancer with brain metastases. A diagnosis of hemorrhagic stroke complicated by posterior anaplastic meningioma World Health Organization (WHO) grade III with pulmonary metastases was confirmed based on the final pathological results.2We report the case of a 58-year-old man who was admitted after experiencing a persistent, severe headache for 5 h, and two episodes of vomiting; however, no further symptoms or coffee-like substances were detected. He had a Glasgow Coma Scale (GCS)  score ofAn emergency department CT scan of the head revealed cerebral hemorrhage in the right cerebellar hemisphere and vermis . The resThe patient underwent right hemisphere tumor resection and external ventricular drainage because of a headache that progressively worsened throughout hospitalization, and a change in consciousness (from lucid to lethargic). We first inserted a right frontal horn external ventricular drain, which allowed us to reduce intracranial pressure during surgery by releasing cerebrospinal fluid and expanding the surgical space, taking into consideration the patient’s gradual increase in intracranial pressure. This prevented the brain tissue from swelling after the skull was opened surgically as well as the development of severe swelling during surgery. Intraoperatively, the tumor appeared fleshy red, mushy, and hypervascular, with peritumoral hematomas. The tumor’s excision size was approximately 4.5 cm × 3.2 cm × 2.5 cm, and there was no clear separation between the tumor and the surrounding brain tissue. Although it was not clear whether the tumor was a high-grade meningioma during the operation, the fact that the tumor was hemorrhagic made us more cautious; we excised the dura that was adherent to the tumor and used an artificial dura to reduce the tension and suture it. We considered that the patient was hemorrhagic from the tumor, and decompression was performed using a debulking flap. Postoperative CT of the head was performed .A malignant mesenchymal brain tumor (right cerebellar mass) was resected, with extremely diverse, and markedly mitotic cells (>20/10 HPF). The immunohistochemical findings were as follows: s-100 (+), a 60% positivity rate of KI-67, H3K27ME3 (+), CD34 (+), Vim (+) and STAT-6(−); the postoperative pathological findings are shown in When comparing the pathology of head and lung cancers, meningioma metastases to the lung should be considered for left lung tumors identified using needle biopsy : Vim(+),Informed consent: Informed consent has been obtained from all individuals included in this study.Ethical approval: The research related to human use complied with all the relevant national regulations, and institutional policies and in accordance with the tenets of the Helsinki Declaration, and has been approved by the Medical Ethics Committee of Yantai Yuhuangding Hospital .33.1The second highest incidence of meningiomas, predominantly composed of arachnoid cells and struts, is observed in the nervous system. Convex and cranial base subarachnoids can arise from various embryonic layers, which explains why meningioma development rates vary from one area of the brain to another. Most studies have reported that intracranial meningiomas are prone to exponential growth ,9,10,11,Meningiomas typically originate on the convex surface of the brain  and thereafter in the sphenoid spine, olfactory groove, stellar region, and posterior cranial fossa at the base of the skull. Epidural and ventricular lesions are rare. Convex surfaces and other non-basal regions have the highest incidence of high-grade meningiomas. Approximately 3–6% of intracranial meningiomas are tentorial ,16. The 3.2MRI and additional CT are usually used to diagnose meningiomas. However, depending on the pathogenic type, meningioma signals on MRI vary. WHO grade I tumors and T2-weighted signals have different associations; grade I tumors are not related to T1-weighted signals. Invasive meningiomas are also more likely to exhibit T2WI hyperintensities , whereasDiagnostic discrimination can be performed in conjunction with diffusion-weighted imaging, magnetic resonance spectrum, and perfusion-weighted imaging for atypical meningiomas ,25,26,273.3WHO grade II and III meningiomas are more likely to show recurrence, aggressive behavior, and distant metastasis . With anA total of 115 patients who had 164 metastases of metastatic meningiomas between 1990 and 2012 were reviewed; approximately 33.9% of the cases were considered to be grade I, 20.9% were considered to be grade II, and 40% were found to be grade III. The most common sites of metastatic disease were the lungs (37.2%), bones (16.5%), intraspinal areas (15.2%), and the liver (9.2%) . Meningi3.4Approximately 1.5–5.4% of all tumors, especially in malignant pathologies such as metastatic neoplasms or gliomas, occur with intracranial hemorrhage . SpontanThese reviews have led to the following current hypotheses regarding hemorrhage ,35,36,373.5We report the case of a 58-year-old man with postoperative pathology confirmed as WHO grade III meningioma complicated by hemorrhage without a supratentorial tumor. The patient’s head MRI results were suggestive of a tumor hemorrhage; we considered metastases and hemangioblastomas for tumors in the posterior cranial fossa and did not consider high-grade meningiomas. In general, meningiomas can be diagnosed using clinical features together with specific MRI findings. However, the probability of meningioma hemorrhage is low, and subsequent concave meningioma hemorrhages are much less frequent. Therefore, we usually treat meningiomas as benign tumors and ignore the meningioma diagnosis.Heavy bleeding and the absence of typical imaging characteristics are more likely to be inappropriately identified as acute cerebrovascular ailments in individuals with HMs who have undergone insufficient imaging examinations . Our patThe characteristics of meningiomas and the side effects of different treatments for different patients vary widely, necessitating individualized therapy. However, the Simpson grade  is stillAdjuvant external beam radiation therapy (EBRT) is very commonly used to treat WHO grade III meningiomas; however, there are no biomarkers to predict the response to therapy, and there is a limited understanding of how to optimize adjuvant EBRT for patients with WHO grade III meningiomas. One week after surgery, the patient was transferred to a rehabilitation hospital. We recommend that the patient undergo radiotherapy at our center as soon as possible; however, the patient chose to undergo radiotherapy at a medical center closer to his home, based on factors such as accessibility and health insurance.If a meningioma metastasizes and recurs, the prognosis is poor, and the patient’s chance of survival is significantly reduced ,42. Loca4Meningiomas are often asymptomatic and any primary symptoms  may often be linked to the effects of a mass. Although meningiomas are typically benign, meningioma hemorrhage and metastatic cases have been documented. Meningiomas may only rarely arise within the posterior fossa; nonetheless, they usually develop along the falx, sphenoid bone, and where there is convexity. Regular follow-up is crucial because of the likelihood of recurrence after meningioma surgery, even if the tumor has been completely resected.


Schwannoma is a benign tumor that originates from the cells of nerve sheaths. The occurrence of mobile schwannoma of the lumbar spine is extremely rare, and when clinicians perform spinal explorations in the expected locations, the results are often negative. This report presents a case of mobile schwannoma in the lumbar spine, aiming to remind doctors to consider the possibility of tumor migration during preoperative planning, thereby avoiding secondary harm to the patient.A 48-year-old male patient presented with a 2-year history of lower back pain and numbness in both lower extremities, which has progressively worsened over the past 3 months. An Magnetic resonance imaging scan revealed an oval-shaped lesion located in the L2/3 vertebral segment, which migrated to the L3/4 vertebral segment following contrast enhancement. The patient underwent tumor resection surgery, and the diagnosis of schwannoma was confirmed through pathological examination.Clinical physicians should increase their understanding of mobile schwannoma of the lumbar spine and utilize imaging techniques to accurately locate the tumor before surgery, in order to avoid unnecessary surgical scope and the risks of additional surgery. Schwannomas are uniformly distributed among various spinal segments and do not show any specific preference for a particular area of the spine. In general, intraspinal Schwannomas rarely metastasize, this article reports a case of a mobile schwannoma of the lumbar spine and provides a systematic review of all cases through PUBMED to improve clinicians understanding of this disease.A Schwannoma, also known as a Schwann cell tumor, is a benign tumor originating from Schwann cells of the nerve sheath. It is commonly seen as a single mass with a complete capsule and represents about 25% of all spinal tumors within the spinal canal.1WI and slightly high signal on T2WI and fat-suppression sequence  of the lumbar spine performed at an outside hospital showed a lesion at the lower border of the L2 vertebral body within the spinal canal. Physical examination revealed slightly hyperactive biceps and triceps tendon reflexes and bilateral Hoffmann signs (+). The lumbar lordosis was flattened, and there was tenderness and right-sided radicular pain upon palpation of the L5/S1 spinous process and paraspinal area, radiating to the right buttock and posterior thigh. Bilateral knee tendon reflexes were reduced, and the dorsiflexion of the foot and ankle muscles was weakened to approximately grade IV . Electrophysiological testing showed evidence of left lower limb radiculopathy involving the L5 root and right anterior tibial muscle. Laboratory tests showed no abnormalities. MRI plain scans showed an elliptical abnormal signal in the spinal canal at the L2/3 level, with slightly high signal on TThrough a posterior midline approach, a tumor resection was performed, along with laminectomy and durotomy at the L3/4 level. The cauda equina was explored, and a spindle-shaped mass was identified with a bundle of nerves passing through the center Fig. C and D.  In 1974, Tomimatsu reported the first case of an intradural-mobile schwannoma, and since then, 29 cases have been reported  years, and 1 case with no reported age and gender. The most common location of the lesion was in the lumbar region (60%), followed by the thoracic region (17%), the thoracolumbar junction (13%), the cervicothoracic junction (7%), and the cervical region (3%). It was observed that all tumors migrated rostral (43%), caudal (43%), or moved back and forth within the spinal canal (13%). The migration distance was within 1 vertebral body in 63% of cases, between 1 and 2 vertebral bodies in 27%, and bigger than 2 vertebral bodies in 10%. One case resulted in entrapment due to migration toward the caudal region after trauma. Clinical symptoms mainly included lower back pain and radiating leg pain with numbness. The activity of the tumor was rarely evident in clinical presentation, and only 3 cases have been reported in the literature, in which symptoms worsened after tumor migration and were related to changes in position or posture, and Valsalva maneuver.,6,9In 1963, Wortzman reported the first case of an intradural-mobile tumor, which was a myxopapillary ependymoma with a distended filum terminale.,14,23,24 In most cases, there are no attachments within the dura mater, and the only restriction for the tumor is the roots connecting the 2 ends. Since there is no spinal cord in the lumbar region, the nerve roots can float in the cerebrospinal fluid, and tumors attached to the redundant cauda equina can have greater mobility. Moreover, when the lumbar spine and buttocks are in an extended position or the weight of the tumor is significant, it can cause the nerve roots to become lax, which can increase the mobility of the attached tumor.,26 Change in posture is also another reason, Isu et al observed tumor activity after gradually changing patient posture and performing repeated MR imaging. Pau et al also observed migrating tumors after changing patient posture following myelography. Therefore, having the patient perform provocative movements, such as back extension, flexion, and jumping, may help detect tumor mobility. Spinal cord deformities caused by extradural tumors may cause corresponding abnormal dilation of the subarachnoid space, 3 cases have been reported in the past, all occurring in the cervical-thoracic region.,11,18 Comparable to the lumbar region, the cervical-thoracic region has a narrower subarachnoid space and shorter nerve roots, so the degree of dilation is often smaller, which may also explain why mobility tumors in the cervical-thoracic region are rarer than in the lumbar region. Some authors have proposed that the Valsalva maneuver can cause tumor movement, and our group has reported 6 cases, all of which moved caudal.,9,13,14 Valsalva maneuver is an action that increases intra-abdominal and intrathoracic pressure by muscle contraction and breath-holding and can be triggered during forceful urination or defecation. The reason why all 6 patients had tumors moving downward may be due to an increase in intra-abdominal pressure causing the tumor to move downward. It has also been reported that the push force of the contrast agent during myelography can cause tumor migration.,6 However, myelography has been replaced by MRI in current preoperative evaluations, and myelography was not used in this case.Several hypotheses have been proposed to explain the reasons for intradural tumor migration, and literature reports have observed redundancy of the cauda equina during myelography.,18 and several authors have reported experiences with second surgeries.,14,17,22 Accurate preoperative localization can reduce the extent of laminectomy and durotomy, as well as the risk of infection and complications for the patient. Currently, various intraoperative or perioperative examinations play a significant role in confirming the precise location of the tumor, such as intraoperative ultrasound, intraoperative MRI, and myelography.,12,13 In the past, myelography was previously considered the gold standard for diagnosis, but the injection of contrast agents can only be completed before opening the dura, and the injection force itself can cause tumor movement, leading to its reduced use. Intraoperative MRI can provide excellent resolution and real-time guidance, but most hospitals do not have this equipment. Friedman confirmed that intraoperative ultrasound helps locate extradural tumors before opening the dura, but it requires higher skills from the operator. Kim suggested that the Valsalva maneuver may help reveal the tumor in situations where there is a lack of imaging modality during surgery.Accurate localization of a tumor before entering the subdural space is crucial for the removal of a mobile intradural tumor, which requires a durotomy and laminectomy. Hollin and Terada reported performing additional laminectomies, with Hollin resection reaching the T12 to L5 level,This study has the following limitations; This study did not conduct long-term follow-up of patients, which limited the evaluation of long-term efficacy and side effects; Patient individual differences in case reports may be large, including age, gender, health status, etc, which may affect the interpretation and promotion of results; Generally speaking, positive or abnormal cases are easier to report and publish, while ordinary or negative cases may be ignored, resulting in publication bias.We reported a case of mobile schwannoma of the lumbar spine, which is a rare disease that clinicians should be aware of. Accurate preoperative diagnosis is particularly important to avoid unnecessary laminectomy and repeated surgeries. Intraoperative MRI, intraoperative ultrasound, myelography, and Valsalva maneuver can all help detect the lesion. When the surgeon does not find the tumor at the site of the dural opening, the possibility of lesion migration should be considered.Data curation: Wanqiu Lyu.Investigation: Xiaojing Li.Supervision: Liyuan Chen.Writing – original draft: Shaowen Song.Writing – review & editing: Binqing Zhang.


Acute abdominopelvic pain in pregnant and postpartum patients presents clinical and therapeutic challenges, often requiring quick and accurate imaging diagnosis. Ultrasound remains the primary imaging investigation. Magnetic resonance imaging (MRI) has been shown to be a powerful diagnostic tool in the setting of acute abdominal pain during pregnancy and puerperium. MRI overcomes some drawbacks of US, avoiding the ionizing radiation exposure of a computed tomography (CT) scan. Although CT is not usually appropriate in pregnant patients, it is crucial in the emergency evaluation of postpartum complications. The aim of this article is to provide radiologists with a thorough familiarity with the common and uncommon pregnancy and puerperium abdominal emergencies by illustrating their imaging appearances. The present first section will review and discuss the imaging findings for acute abdominopelvic pain of obstetric  etiology. Acute abdominopelvic pain, during and just after pregnancy, presents a diagnostic challenge, given the wide range of possible etiologies. For pregnant and postpartum women, the diagnostic approach is often more difficult, owing to several confounding factors. During pregnancy, non-specific leukocytosis, displacement of the abdominal and pelvic organs by the gravid uterus, and difficulty of physical examination can make clinical assessment challenging, especially in the third trimester ,2. On th2 for spatial-peak temporal average intensity ..97].RPOC is defined by intrauterine retention of residual placental or trophoblastic tissue, representing one of the most common causes of post-abortion or postpartum hemorrhage . RPOC coPost-contrast MRI is useful to assess the enhancement, which is also heterogeneous . Other features to take into account are a variable degree of associated myometrial thinning and obliteration of the junctional zone ,103. TheUS plays a key role in patients with suspected GTD to exclude pregnancy as a cause of elevated hCG levels . Once GTUterine AVMs are uncommon and underdiagnosed vascular disorders that may cause life-threatening postpartum vaginal bleeding. AVMs are broadly classified as congenital or acquired. Acquired uterine AVMs usually result from previous trauma, infection, gestational trophoblastic disease, endometriosis, or uterine surgery ,107. CliWhen US is indeterminate, MRI or CT can reveal an ill-defined mass with multiple serpiginous and ectatic vessels in the myometrium and parametrium, connecting to the uterine arteries: this finding, clearly depicted on MR-angiography, corresponds to the hypervascular areas on Color Doppler US, representing a hallmark of uterine AVMs . In congContrast-enhanced CT is the modality of choice for confirmation of the diagnosis in puerperal patients presenting to the emergency department with severe hemorrhage .The definitive diagnosis is still made on the basis of conventional angiography, which remains the gold-standard imaging technique reserved for patients undergoing therapeutic embolization. Arteriography shows a nidus of vessels, supplied by dilated feeding uterine arteries, with high-flow vascular dynamics and early venous drainage .Conservative treatment and observation have been suggested for stable patients with a clinical suspicion of AMV, whereas selective uterine arterial embolization (UAE) or hysterectomy is recommended only for unstable patients. Surgical treatment should be reserved for those women in which UAE is not feasible or contraindicated .Postpartum endometritis is defined as an infection of the decidua or uterine lining that can involve all the layers of the uterus. This complication is the most common cause of low-grade postpartum fever, occurring in 2–3% of vaginal births and in up to 28% of caesarean sections . The likCT and MRI can be employed to further rule out underlying entities such as an abscess or pelvic septic thrombophlebitis.The CT findings of postpartum endometritis are also non-specific, including a thickened endometrium with diffuse wall enhancement and an enlarged endometrial cavity within fluid, air, or debris. MRI can also demonstrate similar findings to CT scans, with air depicted as signal void on T1- and T2-weighted images . ContrasThe treatment of endometritis is broad-spectrum antibiotics administration with proper management for any associated RPOC, infected hematoma or pelvic abscess  113]..113].


This case report describes a diagnosis of spinal pain secondary to metastatic colon cancer, highlighting the need for close monitoring and an interdisciplinary approach for this cohort. An 82-year-old female presented with acute exacerbation of chronic low back pain with a rating of 7/10. She had a history of stage III colon cancer diagnosed 17 years previously and was currently in remission. Red flags included worsening pain after four days of oral analgesics and a history of cancer. Imaging revealed an osteolytic L5 vertebral lesion with endplate disruption, consistent with metastasis. She was urgently referred to an oncologist who ordered chemotherapy and radiotherapy. The metastatic spread of malignancy to the spine can manifest as new or progressive back pain and requires prompt diagnosis and management. Magnetic resonance imaging is recommended for the detection of osseous lesions and spinal cord compression. The case report serves as an educational tool for chiropractors in recognizing and managing spinal metastasis. By sharing this case report, healthcare professionals can learn from the experiences and challenges faced during the patient's care and apply that knowledge to their practice. Low back pain (LBP) is the most common condition that chiropractors treat as primary healthcare practitioners . ChiroprPrompt and accurate diagnosis of spinal metastasis relies upon radiological assessment, with magnetic resonance imaging (MRI) proving to be superior to computed tomography (CT) or radiography for the detection of osseous lesions and cord involvement . ReferraThis report describes a rare case of an elderly female with a history of colon cancer who presented to a chiropractic clinic in Hong Kong, with leg weakness and severe sacral pelvic pain and was diagnosed with metastatic disease of the lumbar spine via advanced imaging. It is crucial for chiropractors to quickly identify patients with metastases and direct them to the appropriate follow-up therapy.An 88-year-old female presented to the chiropractic clinic in Hong Kong, with a 10-day history of lumbosacral pain radiating to the left lower extremity and accompanying paresis, rated 7 on the 0-10 numeric pain scale. She reported a medical history of cerebrovascular disease, essential hypertension, diabetes mellitus type 2, status post-laparoscopic sigmoidectomy for colon carcinoma 17 years ago, and total abdominal hysterectomy with bilateral salpingo-oophorectomy. Current medications included warfarin, metformin, atorvastatin, omeprazole, and aspirin. She denied experiencing headaches, dizziness, or changes in bowel or bladder functions.One month before the presentation, the patient presented to the emergency room with acute left leg paresis. Orthopedic consultations and radiographs of the lumbar spine confirmed the diagnosis of spondylolisthesis and degenerative joint disease Figure . She wasUpon presentation to the chiropractic clinic, the physical evaluation revealed a limited range of motion in the lumbar extension and left lateral flexion that exacerbated the lumbar and sacral pain. Palpation revealed tenderness in the left sacroiliac joint. Neurological examination demonstrated a positive straight leg raise test and a Bonnet test at 45° on the left side. Motor strength in the left L1 to L3 distribution was rated at 4 out of 5 on a numerical scale due to pain. Based on the clinical presentation of left-sided sacral pain, left leg weakness, and neurological findings, the main differential diagnoses were lumbar radiculopathy, lumbar spondylolisthesis, sciatica, a spinal tumor or metastasis, and hip or sacroiliac joint dysfunction. The chiropractor suspected metastasis, especially because of the patient history of cancer, and ordered MRI to probe the structure of the lumbar spine.MRI identified an equivocal lesion in the C5 vertebral body and suspicious osseous lesions in the left ilium Figure , L5 vertPositron emission tomography-computed tomography (PET-CT) revealed a hypermetabolic lung nodule (1.91 x 1.6 cm/SUVmax 15.61), shotty aortopulmonary lymph nodes (0.57 cm/SUVmax 4.55 and 0.67 cm/SUVmax 6.17), and hypermetabolic skeletal lesions on the left ilium (4.47 cm/SUVmax 14.31)  was then begun on the patient and, after six cycles, restaging scans showed a partial response to metastases; however, residual bone pain prompted additional radiotherapy to L5, and this provided significant analgesia. The patient continues close contact and regular follow-up with the medical oncology department and repeats imaging to monitor treatment response and detect disease progression. However, specifics of the colon examination and related findings were not available to the authors, given the limitations of the chiropractic scope of practice.This report describes an elderly female patient in relatively good health who presented with acute exacerbation of a chronic lumbosacral malady to a chiropractic clinic. The condition of the patient was ultimately diagnosed as a recurrent adenocarcinoma of the colon with osseous metastases upon radiological evaluation and confirmation via CT-guided biopsy. Given the patient history of intermittent mechanical low back discomfort, metastatic infiltration of the fifth lumbar vertebra possibly occurred several months prior to the current presentation but remained occult on prior radiographic imaging. However, radiography lacks sensitivity in the detection of neoplastic lesions, with a minimum bone mineral density loss of 30-50% for radiographic visualization .Upon examination, the patient exhibited various warning signs suggestive of serious pathology in addition to her oncologic history, including qualitative changes in symptomatology with pain refractory to analgesic therapy. This warranted further testing via MRI that revealed the underlying etiology of her lumbar symptomatology. The lumbosacral maladies of this patient were most likely secondary to osteolytic destruction and remodeling changes within the L5 vertebral body, consequent to colon cancer metastasis. Despite the presence of two small lumbar disc protrusions, the patient lacked neural foraminal or central canal compromise to substantiate the radicular pain. Sclerotomal discomfort from the L5 metastatic deposit possibly accounted for her clinical presentation. However, as the patient was not fully managed and treated by the chiropractor, identification and monitoring of metastatic lesions on imaging would be beyond the scope of a chiropractor's training and role.Following a search of recent review papers, the Index to Chiropractic Literature, Scopus, PubMed, and Google Scholar, many cases of spinal metastases were found involving diagnosis via chiropractic clinics -25. HoweIn conclusion, this case illustrates the importance of a high level of suspicion for serious pathology in patients with a history of cancer who present with new or worsening spinal pain. Prompt radiological evaluation with MRI is recommended for the optimal visualization of osseous and soft tissue structures. Patients with spinal metastases may present at the chiropractic clinic and require close follow-up and monitoring to assess the therapeutic response and disease progression. A multidisciplinary approach involving the medical and radiation oncologists assigned to the patient may prove most efficacious for the management of pain and optimization of the quality of life in these patients. Future research should aim to establish evidence-based guidelines for the diagnosis and management of spinal metastases, including the evaluation of targeted rehabilitation strategies tailored to patients with comorbid oncologic disease.


Vacuum-assisted excision of breast lesions has come to be widely used in clinicalpractice. Increased acceptance and availability of the procedure, together withthe use of larger needles, has allowed the removal of a greater amount ofsample, substantially reducing the surgical upgrade rate and thus increasing thereliability of the results of the procedure. These characteristics result in thepotential for surgical de-escalation in selected cases and gain strength in ascenario in which the aim is to reduce costs, as well as the rates ofunderestimation and overtreatment, without compromising the quality of patientcare. The objective of this article is to review the technical parameters andcurrent clinical indications for performing vacuum-assisted excision of breastlesions. This results in potential surgicalde-escalation, reducing the extent of the surgical intervention in selected cases,and gains strength in a scenario in which the aim is to reduce costs, as well as therates of underestimation and overtreatment, without compromising the quality ofpatient care.In 1995, vacuum-assisted biopsy (VAB) was introduced as a percutaneous diagnosticmethod for breast lesions and was initially performed with a 14G needle. As of 2010,studies began to report the possibility of excising lesions using this method,either as a secondary benefit or as an initial indication, referring to it asvacuum-assisted excision (VAE). Since then, VAE has been ever more widely used inclinical practice. The greater acceptance and broader availability, together withthe use of larger caliber needles, has allowed the removal of a larger amount ofsample, substantially reducing the rate of diagnostic underestimation and thusincreasing the reliability of the results of the procedureIt is crucial for interventional radiologists to understand the current scenario andthe potential applications of VAE, because it can change the clinical management ofsome breast lesions by updating practices over the years. The objective of thisarticle is to review the technical parameters and current clinical indications forVAE of breast lesions., stating thatthe focus of VAB, the use of which is quite widespread, should be solely ondiagnosis, rather than on removing the lesion completely, whereas VAE, the aim ofwhich is to be a substitute for a diagnostic surgical biopsy and which might not bethe first procedure the patient has undergone, should be focused on removing thelesion in its entirety.The use of VAE begins with the formation of an integrated multidisciplinary team, inwhich the interventional radiologist is responsible for ensuring that the procedureis performed safely and judiciously, under the close supervision of the breastsurgeon, with well-defined eligibility criteria and objectives, and that the excisedsample is evaluated in detail by the pathologist. When performing a vacuum-assistedprocedure, it is important to be explicit about the purpose of the procedure inquestion. The United Kingdom’s National Health Service recommends using specificcodes and consent forms for the various types of vacuum-assistedprocedures argued that there should be no sizelimit for excision, which also depends on the location of the lesion, itsrelationship with adjacent structures, patient comfort, and the risk ofcomplications related to VAE for lesions larger than 3.0 cm. Benign nodules thatshow growth can be submitted to VAE , as well as on that of exeresisas a minimally invasive alternative to conventional surgery for small, incipient,biologically favorable tumors detected by screening,6.To date, vacuum excision has had numerous applications: for excision of previouslybiopsied lesions with a histological diagnosis of high risk or uncertain malignantpotential (B3 lesions); for exeresis of benign lesions when the procedure has beenrequested by a clinician; and for a repeat biopsy in cases in which there isdiscordance between the radiological and pathological findings. The potentialapplications of VAE have been described in recent studies. For example, one studydescribed the use of VAE for the excision of gynecomastia. The authors concluded that greaterutilization of VAE in selected cases could reduce health costs and avoid unnecessarysurgical procedures.In a study comparing the cost of VAE with that of open surgery, VAE was found to bethe less costly of the two, for benign lesions and for high-risk lesions, even whenthe cost of the follow-up imaging evaluations was taken into account. Because it allows a definitive diagnosis ofcancer to be made in patients scheduled to undergo surgery for conditions otherthan cancer, VAE can play a diagnostic role by identifying malignancy in thepreoperative period, thus ensuring the correct surgical management of such casesin a single procedure. Data in the literature underscore the need for thealternative management of B3 lesions and suggest that VAE is appropriate inselected cases,4,7-11. For VAE ofhigh-risk breast lesions, the United Kingdom’s National Health Servicerecommends obtaining 4 g of material, the number of fragments depending on thecaliber of the needle used,as detailed in High-risk lesions, which are detected in 4-9% of biopsies, constitute aheterogeneous group of lesions with uncertain malignant potential, given therisk of underestimation in the surgical upgrade, and can be precursor lesionswith the potential to progress to invasive carcinomasuggested that radial scarring without atypia and papillary lesions withoutatypia should be treated by vacuum excision, and that, in cases in which VAE wasperformed after VAB, it was mandatory to identify, histologically, the center ofthe previous biopsy and the reaction to the clip, in order to have a reliablecorrelation with the upgrade of the lesion removed. The authors stated that,based on the specimen fragments sampled, the pathologist could not comment onwhether there was complete excision of the lesion and its margins, theconfirmation of which would depend on the radiological impression. However, inclinical practice, some groups have tried to overcome this limitation by sendingthe material to pathology in separate vials, one with the fragments that containthe lesion and one with the fragments that include the margins removed in a360-degree resection around the lesion. Studies have shown that VAE is safe incases of papillary lesions without atypia, presenting a rate of upgrade tocarcinoma of 0%, compared with approximately 10% for core biopsy,13.In 2019, Shaaban et al.. In 2021, Catanzariti et al. suggested that VAE could be used in cases ofpapillary lesions without atypia; radial scarring with or without atypia; flatepithelial atypia; and lobular neoplasia . However, for casesof atypical intraductal epithelial proliferation, the authors suggested that VAEbe used only in cases of small lesions with atypical ductal hyperplasia that isunifocal.Since the first international consensus conference on lesions of uncertainmalignant potential (B3 lesions), in 2016, to the present day, the approaches tosuch lesions have been constantly readjustedinsitu, and residual microcalcifications should always be excisedafter the procedure.When there is lesion present in the margins evaluated, it is important todetermine the radiological-pathological correlation. In the case of flatepithelial atypia, given its mild relative risk-1 to 1.5 times greater than thatof breast cancer, similar to that attributed to typical usual ductalhyperplasia-there is no recommendation in the literature for tumor-free marginsif there are no other lesions with significant pathology, such as atypicalductal hyperplasia and atypical lobular hyperplasia/lobular carcinoma ,4: if it is a benign lesion without atypia, the patientis referred for regular follow-up; if it is a lesion with focal atypia, thepatient is followed, under active surveillance of the biopsied region, for aperiod of five years, including evaluation by imaging methods, such as magneticresonance imaging, in selected cases; and if it is a malignant lesion, thepatient is referred for definitive, therapeutic surgery, diagnostic surgerytherefore being precluded. To select the patients to be followed, each case mustbe assessed for commitment to follow-up, taking into account factors such as the age and risklevel of the patient; the size of the lesion and its relation to the volumeremoved; whether there is residual lesion; and whether the lesion in questionwas a primary or incidental histological finding.The United Kingdom’s National Health Service guidelines for the management of B3lesions state that, in order to adopt the appropriate management, thosesubmitted to core biopsy, fine needle aspiration, or first-line VAB can bereferred for VAE as a second-line procedure. In such cases, the followingprotocol is followed, depending on the histological diagnosisin situ;extensive lobular carcinoma in situ with necrosis oraccompanied by other high-risk lesions; the combination of flat epithelialatypia and atypical ductal hyperplasia; and palpable papillary lesion withatypia, papillary flow, and calcifications. On the basis of the currentknowledge, VAE can be indicated in cases of papillary lesion without atypia,radial scarring, flat epithelial atypia, unifocal atypical ductal hyperplasia,and classical atypical lobular hyperplasia/lobular carcinoma insitu, especially when identified as incidental findings.It should be borne in mind that surgery is still the rule in cases of atypicalductal hyperplasia; pleomorphic lobular carcinoma :hospital admission; induction of anesthesia, medical and hospital materials;preoperative localization; and considerable time spent in preoperative visits.In addition, the postprocedure aesthetics and the prolonged recovery time afteropen surgery result in lower patient satisfaction. The main clinical indications for VAE arelow adherence to follow-up, patient anxiety, increase in lesion volume duringfollow-up, symptoms, and planning to start hormone replacement therapy forassisted reproduction,17.For the vacuum excision of benign lesions, VAE has the advantage of greateraffordability in comparison with the surgical procedure, which would involve thefollowing.In addition, the rate of recurrence is lower for lesions smaller than 1.5 cm,making VAE an alternative to surgery for the complete excision of benign tumors.In cases in which the results of previous biopsies were discordant, it can beadvantageous to use VAE because it allows the entire lesion to be aspirated,thus avoiding a lesion sampling error, as often occurs in complex solid-cysticlesions. In cases of mild gynecomastia, not requiring surgical reduction of theskin or of the nipple-areolar complex, VAE is considered a viable alternative tothe classic surgical procedure for gynecomastia, resulting in smaller scars andshorter hospital stays, with similar aesthetic results,20.For nodules that show rapid growth, such as phyllodes tumors, studies show thatthe post-VAE recurrence rate is 5-17% in benign cases when 3.3 cm is used as thelesion size cutoff point,6, one notable effort is the RESPONDER clinicaltrial, which wasdesigned to evaluate the accuracy of VAB for the diagnosis of a pathologicalcomplete response-defined as the absence of residual lesion in the tumorbed-after preoperative neoadjuvant treatment, given that, depending on themolecular subtype, a pathological complete response can be achieved in up to 60%of patients with breast cancer. However, the accuracy of imaging evaluationafter neoadjuvant treatment can be limited and surgery is therefore consideredmandatory at the end of treatment, in order to remove residual disease orhistologically diagnose the treatment response. Since the 2018 announcement ofthat trial, no results have been published, because of problems with patientrecruitment and the difficulties provoked by the pandemic. The authors pointedout that the histopathological evaluation of a non-tumor sample is a criticalissue, creating uncertainty as to whether the region of the tumor with apathological complete response has been sampled or if unrepresentative tissuewas removed, which could introduce a sampling error. Similarly, the SMALL clinical trial wasinitiated in the context of a discussion about incipient, biologically favorablecancers diagnosed by screening and whether they could be treated by VAE, thusprecluding surgery as well as minimizing overtreatment, morbidity andcosts. The authorsapplied the following eligibility criteria: being over 47 years of age; havinghad no previous breast tumor; and currently having a tumor that is smaller than1.5 cm, contains no microcalcifications, is unifocal, is classified as grade 1,is strongly positive for estrogen/progesterone receptors, is negative for humanepidermal growth factor receptor 2, and does not involve axillary lymphnodes.Among the ongoing studies of the potential applications of VAEAfter surgical excision in patients submitted to VAE with a result of malignancy,the major axis of the infiltrative neoplasm-in the biopsy specimen and in thesurgical specimen-should be measured on its longest axis and those data shouldbe correlated with the prebiopsy imaging, in order to determine the size of thetumor for staging. If the tumor is small and there is no residual lesion in theresected specimen, the tumor-node-metastasis staging should consider thedimensions of the tumor as measured on the prebiopsy imaging, because of thefragmentation of the lesion, and it is important to send a detailed report withthe correct documentation of the lesion and measurements before the procedure isperformed .-24.Post-VAE complications, which have been observed in various interventional breastprocedures, include ecchymosis, infection, pseudoaneurysm, and pneumothorax, as wellas others related to the vacuum procedure, such as hematoma, skin laceration, clipmigration, scarring, postbiopsy distortion, and fat necrosis, with or withoutcalcification. It is extremely important to know not only how to perform theprocedure but also how to manage complications promptly, accurately, and resolutely,which is the responsibility of those who perform the procedure. Hematomas constitutethe most common complication and can predispose to clip migration, together withrapid breast decompression. Skin laceration in the region can be avoided by usingthe Berná-Serna maneuver, which consists of fixing a cannula between the skinand the lesion, in order to prevent the skin from being suctioned when the vacuumsystem is activatedThe applicability of VAE relies on a multidisciplinary approach, with closecommunication between members of the multidisciplinary team regarding when toindicate the procedure, which complications are acceptable, what are the bestpractices after the procedure, and which follow-up regimen should be employed. Inrecent years, the technique has evolved considerably. Notably, the pathologicalresults have improved, approaching those of sectorectomy/nodulectomy analyses, withthe use of the information and measurements that are necessary for ensuring thereliability of the method. Such advances have led to greater popularization of VAE,resulting in surgical de-escalation in properly selected cases. However,interdisciplinary integration and synergy are vital for the success of themethod.


A new biliary duct (BD) stricture raises questions about the presence of malignancy, especially with a history of metastatic pancreatic cancer. A few cases of colloid carcinoma (CC) of the pancreas have been published, but none have described recurrence in the biliary tract. We report a case of intrahepatic biliary CC that recurred after two years after the last dose of immunotherapy for pancreatic CC. In addition to a unique biliary cancer case presentation, this case raises awareness of the best strategy for cancer surveillance. Colloid carcinoma (CC) of the pancreas is a rare variant of ductal adenocarcinoma (DAC) and represents 1% of pancreatic carcinomas . CC is aThis article was previously presented as a meeting abstract at the ACG Annual Scientific Meeting on October 25, 2020.A 72-year-old female with a history of stage IV pancreatic CC with metastases to the liver, spleen, and stomach presented with dark urine and jaundice. Diagnosed five years earlier, she required distal pancreatectomy, splenectomy, and partial gastrectomy. At that time, pathology showed poorly differentiated CC of the pancreas, invading the spleen and stomach with negative surgical margins. Unfortunately, one month later, metastatic liver lesions were found during post-surgical follow-up. She failed gemcitabine, paclitaxel, liver CT-guided microwave ablation, and a combination of leucovorin calcium, fluorouracil, and oxaliplatin therapy. Pembrolizumab was started for progression and showed stable disease for two years but was stopped due to neutropenia. Two years after the last dose of immunotherapy, magnetic resonance cholangiopancreatography (MRCP) for surveillance showed new moderate left intrahepatic duct dilation with left hepatic duct narrowing Figure . She underwent endoscopic retrograde cholangiopancreatography (ERCP) twice, requiring biliary stent placement and brush cytology with benign results. Discussion at the multidisciplinary tumor board led to a consensus on the intraductal papillary mucinous neoplasm of the bile duct (IPMN-B). During the most recent hospitalization, labs showed elevated liver function studies with a total bilirubin of 4 mg/dl. ERCP was done with cholangioscopy, demonstrating a large frond-like mass partially obstructing the left main hepatic duct Figures -3. It was biopsied, and pathology was reported as CC.CC of the pancreas is a sub-category of DAC differentiated by histopathology, and at least half of the epithelial cells are mucinous components comprising the tumor . The mucClinical presentations in CC of the pancreas are often non-specific. Patients can present with nausea, vomiting, abdominal pain, back pain, weight loss, or anorexia. In cases where the tumor obstructs the main pancreatic duct, the patient can experience pancreatic insufficiency and malabsorption of nutrients . HoweverThe diagnostic approach typically begins with imaging, where MRI and CT scans are less invasive and highly useful prior to surgery. MRCP can be helpful to recognize BD strictures and dilation of the ducts. As in our case, the presence of a new stricture raises concerns about malignancy. Performing ERCP with cholangioscopy can be particularly helpful in identifying lesions and obtaining more accurate pathology samples. A frond-like mass on BD under cholangioscopy raises suspicion for malignancy and warrants a direct lesion biopsy. Endoscopic ultrasound with a fine-needle aspiration can also aid in confirming malignancy ,11. It iIn conclusion, our case highlights the challenges in distinguishing between IPMN-B and CC of the BD. A new BD stricture raised concerns about the presence of malignancy, especially given the patient's history of metastatic pancreatic cancer. The cholangioscopy findings and the pathology report should make us investigate the best strategy for cancer surveillance since brush cytology can often yield negative results. Further studies are warranted to explore the recurrence and metastasis rates in pancreatic CC.


Developing in a limited space, rare tumors located at the nose and paranasal sinuses are sometimes difficult to diagnose due to their modest clinical presentation, which is uncorrelated with anatomopathological diversity. This limits the preoperative diagnosis without added immune histochemical study; for that reason, we present our experience with these tumors with the intention of raising awareness. The patient included in our study was investigated by our department through clinical and endoscopic examination, imaging investigations, and an anatomic-pathological study. The selected patient gave consent for participation and inclusion in this research study in compliance with the 1964 Declaration of Helsinki. Tumors in the nose and paranasal sinuses are rare lesions, affecting <1 in 100,000 people per year [While new discoveries are made worldwide, especially to assess new methods of prevention, diagnosis, or specialized treatment, the rarity of these cases remains an obstacle in choosing the management option.Olfactory neuroblastoma, also referred to as esthesioneuroblastoma, represents an oncological entity of neuroectodermal origin arising in the upper part of the nasal cavity. Its incidence represents 2–3% of all nasal neoplasms [The etiology and risk factors are still unknown. As with most sinonasal tumors, the symptoms are nonspecific, most commonly epistaxis, nasal obstruction, and hyposmia. A biopsy is essential for diagnosis, while CT scans and MRI images are used for the staging system.These tumors are locally aggressive, with the propensity to spread into the anterior skull base as well as to metastasize to the cervical lymph nodes, thorax, and bones .Cervical metastases are described in 5–8% of cases at diagnosis and in 15–25% of patients as recurrence ,5. The mIn our day, three separate staging systems exist. Kadish et al.  proposedThere is no agreed-upon standard treatment for olfactory neuroblastoma. Surgical treatment and radiotherapy represent the most frequently used approaches. Chemotherapy treatment modalities can be used in selected cases. Open surgical approaches, such as extracranial and anterior craniofacial resection, are preferred in the treatment of ONB. The development of an endoscopic approach over the past few decades has gained popularity and offered many advantages , representing a valid treatment for olfactory neuroblastoma ,8.Multimodal treatment associating surgery with radiation therapy has been demonstrated to have the best survival rates; however, the infrequency of olfactory neuroblastoma and its heterogeneous clinical biology limit the possibility of creating specific protocols of treatment ,9,10,11.Recurrence may be encountered years after treatment; therefore, long-term follow-up is recommended .Multiple myeloma currently affects 250,000 people globally . AccordiThe diagnosis is made based on the evidence of one or more of the CRAB criteria , with biopsy confirmation of bone marrow infiltration by 10% clonal plasma cells or detection of a plasmacytoma. The laboratory diagnosis includes a variety of biochemical examinations  associated with the monitoring of end-organ damage ,15.A 45-year-old patient was admitted to our department with epistaxis from the right nasal fossa. He received conservative treatment using an anterior nasal tampon (Merocel) for 2 days, and he returned a few days later with recurrent symptomatology. The patient underwent an endoscopic examination, where he presented a solitary, smooth mass in the right nasal fossa.The patient underwent CT and MRI of the head and neck prior to surgical resection; the CT scan identified a heterogeneous, natively hyperdense tissue mass, located at the level of the right nasal cavity, which it filled for the most part. The mass-produced remodeling and bone erosion, with partial visualization of the right inferior nasal turbinates. The native CT appearance was nonspecific in the differential diagnosis, including an inverted papilloma, sinonasal polyp, or adenocarcinoma. The right maxillary sinus was filled with mixed, parafluid, and tissue densities .The patient underwent a chest and abdominal CT scan and MRI, and there was no proof of distant metastasis. The patient was staged according to the Kadish system.The tumor was located in the nasal fossa, without intracranial extension or erosion of the cribriform plate. An endoscopic approach was performed. The tumor was successfully removed with negative margins; a right maxillary antrostomy was performed. A Merocel was placed in the nasal fossa and removed after 48 h. In order to avoid the risk of infection, for all endoscopic surgeries, we use an antibioprophylaxis for 48–72 h, initiated on the day of the surgery.The patient had no complications , and no epistaxis was found after the postoperative Merocel removal. The patient stayed in the hospital for less than a week.Tissue fragments were immunohistologically studied; the fragments were initially fixed in formalin. Positive and negative controls were performed.The tumor consisted of a polypoid thickened respiratory mucosa through a pseudolobular tumor proliferation that develops exclusively in the lamina propria, respecting the structure of the covering epithelium. Tumor cells were relatively monomorphic, small in size, weakly basophilic or weakly acidophilic, with a nucleus with irregularly or finely dispersed chromatin and inconstant nucleoli; some cells have a central nucleus, and in other cells, the position of the nucleus is eccentric, rare mitoses (1–2 cp × 40), with solid and trabecular growth architecture in a fibrous stroma and the formation of pseudorosettes (Homer Wright rosettes), and frequently in perivascular position, encasing small and medium vessels.Positive and negative controls were performed.Immunohistochemical reactions are performed on the paraffin block. The negative immunoreactions in tumor cells were identified . BCL-2 was positive, and PGP9.5 was positive.Correlating the histological aspects  with immAfter the surgical endoscopic removal of the tumor and the confirmation of the diagnosis by the anatomopathological exam, the patient was referred for radiotherapy. He underwent 33 sessions of radiotherapy treatment (DT = 50 Gy/fr/38 days) on the right nasal fossa. Radiotherapy was well tolerated.Follow-up was conducted by the oncology and radiotherapy departments, where postoperative imaging of the head and neck was performed. No recurrence was identified. The ENT follow-up was scheduled every 3 months during the first year for nasal endoscopy, every 6 months during the second and third years, and annually thereafter without recurrence or symptoms.In the fifth year after the primary diagnosis, the patient complained of lumbosacral and humeral bone pain.He underwent a CT scan and an MRI, which indicated osteolytic lesions of the vertebral bodies C6, C7, C8, T7 (16 mm), and T11, as well as the costal arches and bone basin (11 cm at the level of the right iliac wing with the extension to the right iliac muscle and a 10 mm lesion at the left iliac wing). The CT scan identified an important circumferential thickening of the mucosa of the right maxillary sinus that almost completely occupied it, with otherwise normally aerated paranasal sinuses.The lesions raised suspicion of multiple myeloma or long-distance metastasis. A biopsy of the sinus mucosa was performed with local anesthesia; the patient was referred to a hematologist and scheduled for a whole-body positron emission computer tomography (PET-CT) and a bone biopsy. The sinus biopsy revealed hypertrophic mucosa and the absence of tumoral cells.The whole-body PET-CT showed the activated metabolism of a mass in the right iliac wing measuring 10 to 7.5 cm, which invaded the neighboring endo- and exopelvic structures. The mass showed an inhomogeneous uptake of FDG. Other similarly moderate FDG-capturing osteolytic lesions could be distinguished in the right posterior fourth costal arch , the right fourth anterior costal arch, the left sixth lateral costal arch, the sternal manubrium, the medial angle of the left scapula, C7 (with a major risk of subsidence), T7, and T9 vertebral bodies, the right lateral clavicular extremity, the right humeral head, apex of the right temporal bone, the right parietal bone, and the left sciatic tuberosity .The bone marrow biopsy concluded medullary iron blockage with moderate hyperplasia of the plasmacyte, affecting 4.5% of nucleated cells.Based on the complete blood cell counts, the protein electrophoresis, the serum electrophoresis, the immunoquantification, and the bone marrow biopsy, the diagnosis of monoclonal gammopathy of unspecified etiology of type IgG with lambda chains was established.A bone biopsy was taken from the iliac lesion, demonstrating the diagnosis of multiple myeloma type IgG with kappa chains . The Ki37 cell proliferation index was 40%. The disease was classified as stage III according to the International Staging System (R2-ISS) for overall survival in multiple myeloma.The patient underwent hematological treatment with daratumumab, bortezomib, thalidomide, dexamethasone, and bisphosphonate. The symptoms of bone pain were relieved. The patient is still in the follow-up stage.As a rare malignant tumor of the nasal cavity with a frequency of only 0.4 million per year, olfactory neuroblastoma was described for the first time in 1924 .The tumor can affect both children and adults; in adults, the disease generally occurs between the fifth and sixth decades of life. Its origin is still unknown. No lifestyle risk, environmental, or geographic factors are linked to its apparition. The symptoms may vary from unilateral nasal obstruction 70%) to epistaxis . The goHaving neuroectodermal and epithelial origins, olfactory neuroblastoma presents itself as a unilateral, polypoid tumor formation of low consistency with a nonspecific clinical presentation. The olfactory epithelium can be identified in the mucosa of the superior and middle turbinates and also in the mucosa of the nasal septum .The immunohistochemicalexamination is based on positive markers for S100, BCL2 , and PGPOlfactory neuroblastoma poses a high risk of local invasion, recurrence, and distant metastasis . DulguerCastelnuovo et al. reported, in a total of 10 patients treated with endoscopic surgery, the presence of cervical metastasis 21 months after surgery. The patients underwent a bilateral modified neck dissection plus radiotherapy . Some stBased on our experience, we think that the endonasal approach achieves a complete resection for small, localized lesions when no reconstruction is needed, and all the lesions can be resected with negative margins. Our patient was, at the time of the diagnosis, N0M0, so we did not perform a neck dissection. The endonasal excision allows rapid recovery and returnsto daily activities for the patients, improving their quality of life.The infrequency of these tumors has limited the possibility of categorizing the prognostic factors and specific protocols of treatment. Several staging systems have been proposed. The most commonly used was proposed by Kadish et al. in 1976 and modified in 1993. Nowadays, some institutions apply the TNM staging system by the AJCC based on the Dulguerov modified version of staging ,21 Tabl.A meta-analysis compared the outcomes of the Kadish and Dulguerov staging systems, finding that both systems correlated with prognostic factors in terms of disease-free and overall survival, with the Dulguerov system having a superior performance .CT and MRI images are essential for correct staging. PET-CT can identify local recurrences and metastases. The anatomopathologicalexamination is the gold standard for diagnosis.The typical recommended treatment for olfactory neuroblastoma consists of endoscopic surgery resection associated with radiochemotherapy. Endonasal endoscopic surgery is preferred due to its efficient local control and lower morbidity . In our However, in this particular case, the imaging suggests no involvement of the cribriform plate, base of the skull, orbit, or intracranial cavity. As described in the literature, endoscopic resection allows the total excision of small lesions. Due to its location in the right nasal fossa and the fact that it had not spread into the adjacent structures, we performed an endoscopic approach.In a retrospective study by Gallia et al., eight patients with olfactory neuroblastoma treated by endonasal endoscopic surgery were identified. They had a complete resection and negative intraoperative margins, with no evidence of disease over a mean follow-up of over 27 months .Newer radiotherapy techniques have been added, reducing cerebral and ocular toxicity over time . NeoadjuDue to the delayed regional recurrences associated with olfactory neuroblastoma, prolonged surveillance is recommended. We highlight in our report the aggressiveness of this tumor and the importance of including PET-CT in the monitoring follow-up protocol for olfactory neuroblastoma while also considering that distant metastases (approximately 10%) can occur irrespective of the grade of the tumor .In our report, osteolytic lesions raised the suspicion of distant metastases of the olfactory neuroblastoma and multiple myeloma. Distant recurrences of olfactory neuroblastoma are described in the literature. In an article by Loy et al., 34% of patients developed recurrent disease, and the most distant metastases were osseous  . The symIn multiple myeloma, malignant plasma cells proliferate in the bone marrow, displacing normal blood cells and leading to disease and symptom manifestations such as generalized weakness, weight loss, bone pain, hypercalcemia, and anemia .Although our patient was diagnosed with a hematological disease and there is no correlation between multiple myeloma and olfactory neuroblastoma, it should be noted that long-term follow-up of an olfactory neuroblastoma patient is mandatory. The presence of a multidisciplinary team around the patient can identify and treat relapses, long-distance metastases, or even a hematological disease.Rare sinonasal tumors present similar symptomatology, originating in a relatively small anatomical space. The patients often have a long history before their initial presentation. The treatment modalities have changed over time with the evolution of endoscopic surgery, and a multidisciplinary approach may improve the survival rate and the patient’s quality of life. Lifelong follow-up is crucial, combined with imaging surveillance, given the possibility of distant metastases occurring many years after treatment of the primary tumor or, as in our case, an early diagnosis of hematological disease.