CTSK gene, c.953G > A, p. (Cys318Tyr). The proband had a good postsurgical response with sustained improvement in his sleep OSA. We present here the history and clinical characteristics of a patient with typical features of pycnodysostosis and an unusual presentation and histopathology of gnathic bone lesions. This report adds to the body of literature on this rare condition and also highlights the finding of giant cell-rich lesions of the gnathic bones. Giant cell-rich lesions in pycnodysostosis have previously been reported in two cases in the literature. While there is not enough evidence to support a certain association with pycnodysostosis, it is prudent to consider regular oral dental reviews in affected individuals to identify pathology early and avoid such life-threatening complications.Pycnodysostosis is a skeletal dysplasia characterized by short stature, generalized osteosclerosis, acro-osteolysis, and recognizable facial features. Oral manifestations are commonly described and include a high-arched palate with dental crowding and malocclusion, hypoplastic enamel, and retained deciduous teeth with impacted permanent teeth, and there is an increased risk of developing osteomyelitis of the jaw. We report here the history of a 9-year-old male with the typical facial and skeletal phenotype of pycnodysostosis but novel oral features. He presented with bilateral progressive facial swelling, which caused functional impairment with chewing and contributed to his severe obstructive sleep apnea (OSA). The severity of his OSA required surgical intervention, and the lesions were resected. Extensive bone remodeling and replacement by fibrous tissue were noted on submucosal dissection, and bilateral subtotal maxillectomies were required. The histopathology of the biopsied lesion was consistent with a giant cell-rich lesion. Genetic testing identified a pathogenic homozygous variant in the  Pycnodysostosis OMIM #265800) is a skeletal dysplasia classified as an osteopetrosis condition . It is c65800 is Oral manifestations are commonly described and include a high-arched palate with dental crowding and malocclusion, hypoplastic enamel, and retained deciduous teeth with impacted permanent teeth, and there is an increased risk of developing osteomyelitis of the jaw , 4. OtheCTSK gene. This gene encodes cathepsin K, which produces a lysosomal cysteine protease that is highly expressed in osteoclasts and plays a role during bone resorption and remodeling. Loss of function variants are known to be causative in pycnodysostosis. The osteosclerotic phenotype is explained by the lack of lysosomal cysteine protease, while osteolysis is thought to be due to a site-specific variation in the enzyme action causing increased bone resorption  with bilateral, progressively enlarging cheek masses, airway obstruction, and difficulty chewing food.He was born to his non-consanguineous parents at term after an uneventful pregnancy with normal birth growth parameters; his postnatal course was uncomplicated, with no early failure to thrive. At 1 year of age, he presented with upper airway obstruction initially conservatively managed, but he later underwent an adenotonsillectomy at 3 years. Despite this, his upper airway obstructive symptoms remained a concern. At 8 years of age, he was noted to have swelling of both cheeks, which progressively worsened.At a presentation at 9 years, the proband's growth parameters showed height and weight below the 3rd centile and a head circumference on the 25th centile. His facial features were striking for asymmetrically swollen cheeks. Additional clinical findings included bifrontal narrowing with frontal prominence, proptosis, a flattened nasal bridge with a convex nasal profile, a long philtrum, and retrognathia ,B. He haThe right-sided facial mass was approximately 3 cm × 4 cm, with multiple smaller mobile lesions that were not tender. The left-sided lesion was smaller, measuring 2 cm × 1 cm, also with smaller mobile lesions, some of which were tender. Oral examination showed prominent dental decay, over-retained primary teeth, impacted permanent teeth with displacement and overcrowding, and intraoral extension of the mass lesions . The palSeveral special investigations were conducted; skeletal x-rays showed generalized sclerosis with acro-osteolysis, and a CT scan of his head revealed expansile mixed-density masses involving the maxillae and mandible bilaterally . The manThe multifactorial OSA was severe, and a decision was made to intervene surgically and resect the lesions. During surgery, the otorhinolaryngology and maxilla-facial team secured the airway with a tracheostomy and proceeded to resect the lesions via an oral approach and vestibular incision. Extensive bone remodeling and replacement by fibrous tissue were noted on submucosal dissection, and bilateral subtotal maxillectomies were required. The mandibular lesion was not removed, as further spontaneous regression was expected. Two weeks later, the patient returned to the theatre for impressions and obturator construction, which was subsequently inserted when the tracheostomy was decannulated. The proband showed a good response with marked improvement in his OSA postsurgery.The histopathology of the biopsied lesion showed aggregates of multinucleated osteoclast-type giant cells with admixed mononuclear spindle cells and intervening fibrous bands . MitoticCTSK gene, NM_000396.3:c.953G > A, p. (Cys318Tyr). Notably, no pathogenic or uncertain variants were found in SH3PB2, the gene responsible for cherubism initially suspected in the proband. The diagnosis of pycnodysostosis was confirmed, and the family and proband counseled accordingly. Genetic testing of the tumor tissue was not undertaken.Genetic testing was undertaken in the proband with a commercially available next-generation sequencing osteogenesis and bone fragility panel . The result showed a pathogenic homozygous variant of the We present the history and clinical course of a Zimbabwean child with pycnodysostosis who presented with bilateral giant cell-rich lesions of his gnathic bones. While the clinical features were typical of pycnodysostosis, the oral manifestations present in this child have not been as well delineated. This highlights the need for regular and careful oral and dental reviews in this rare condition.S3HBP2. In a few cases, despite a clinical diagnosis, a molecular cause is not found, suggesting possible genetic heterogeneity , which does not share a common protein domain with the current patient's variant in exon 8. In addition, only the first patient reported a traumatic event preceding the development of the giant cell-rich lesion. With fewer than 500 cases of pycnodysostosis reported (The oral manifestations in pycnodysostosis previously described include dental crowding, a grooved and high-arched palate, malocclusion of teeth with hypoplastic enamel, hypercementosis, and often retained deciduous teeth with impacted permanent teeth. One of the important complications facing patients with pycnodysostosis is osteomyelitis . The lesreported .CTSK variants associated with pycnodysostosis (There are more than 35 sostosis ; the prosostosis , 11. Girsostosis , 11. TheTo summarize, we present here the history and clinical characteristics of a patient with typical features of pycnodysostosis but an unusual presentation and histopathology of gnathic bone lesions. This report adds to the literature on this rare condition and also highlights the finding of giant cell-rich lesions of the gnathic bones. While there is not enough evidence to support a certain association with pycnodysostosis, it is prudent to consider regular oral dental reviews in affected individuals to identify pathology early and avoid such life-threatening complications.


We report a rare case of the continuous type of splenogonadal fusion (SGF) in a young adolescent with preserved testis.A 19-year-old male patient with a history of left inguinal hernia repair 10 years ago presented with a palpable mass on the left side. Computed tomography revealed a 58x37mm mass with a tissue density of 47HU, demonstrating vigorous enhancement following contrast administration and displaying well-defined margins with the left testicle. It was noted to be growing vertically in the left inguinal canal and to be continuous with the lower pole of the native spleen. The patient underwent laparoscopic surgery to remove the splenic tail in the abdomen and to separate the scrotal spleen from the left testicle through the left inguinal tract. The histopathological examination confirmed the presence of splenic tissue.SGF is often diagnosed incidentally during exploration or surgery for scrotal swelling or mass, cryptorchidism, or inguinal hernia in young patients. It is important to be aware of this condition to avoid unnecessary radical orchiectomy.Diagnosing the SGF preoperatively can be challenging. However, a combination of imaging modalities and negative tests for alpha-fetoprotein (AFP), lactate dehydrogenase (LDH), and beta-human chorionic gonadotropin (b-HCG) can aid in making an initial diagnosis. The use of laparoscopic surgery can further improve the diagnostic process, allowing clinicians to accurately diagnose SGF and make well-informed treatment decisions. •Multi-scan computed tomography showed a left scrotal mass continuous to the lower pole of the native spleen which is typical of SGF.•Laparoscopy revealed a typical appearance of the splenic tail, further strengthening the diagnosis of SGF.•Case report of the continuous type of SGF, the first of its kind in Vietnam in the English literature. SGF m22.1A 19-year-old male patient, who is a university student and resides in Thai Binh province, Vietnam, has an average family income.2.2The patient noticed a mass in the left scrotum for the past 3 months, which increased in size and did not cause any pain. Regarding his past medical history, he was a healthy-term baby with a previous history of a left inguinal hernia. He underwent open surgery at ten years old, during which this defect was not detected.Clinical examination showed that the patient was in average physical condition with a BMI of 20. The external shape of the penis was normal, and the pubic hair was at Tanner stage 4. The left testicle can be palpated and the mass firmly attached right next to the left testicle was 4x3cm in size, firm, with clear boundaries, gradually tapering to the external inguinal ring A.Fig. 1C2.3First, during the ultrasound, we observed a mass located next to the left testicle. The mass had a volume of 20 cm3 and appeared to have a similar structure to testicular tissue. However, it was hypoechoic and mildly hypervascular. Based on these findings, my sonographer suspected a third testicle was present in the scrotum. Additionally, we conducted an abdominal ultrasound, chest X-ray, and echocardiography, which did not detect anything abnormal. An assay for testicular tumor markers was requested: AFP, b-HCG, and LDH were found to be normal. Multi-scan computed tomography detected a mass measuring 57x38mm, tissue density (47HU), strong enhancement after injection, mass with clear margins and boundaries pushing the testicles down, growing along the left inguinal canal up to left iliac fossa and continuous with the lower pole of the spleen B. The pr2.4The patient underwent exploratory laparoscopic surgery and it was observed that the malformed spleen grew from the lower pole of the spleen. It followed the colonic groove and entered the inguinal canal, ultimately sticking to the testicles. At this time, the diagnosis of SGF was confirmed. We performed a resection of the splenic tail in the abdomen A and surThe histopathological examination confirmed the presence of splenic tissue.2.5After surgery, the patient was stable, had no short-term complications such as bleeding or infection, and was discharged after 3 days. We schedule patient follow-up visits after 1 month and 6 months to monitor complications such as testicular atrophy, and recurrence, and evaluate the patient's fertility.3SGF is a very rare congenital anomaly and was first documented by Bostroem in 1883 , and an This anomaly concerns almost exclusively the male sex, with a male-to-female ratio of 14–15:1 ,31. HoweThe etiology of SGF remains incompletely understood; however, the prevailing hypothesis posits its occurrence during gestational weeks 5 to 8, before descent of the gonads commences. Between the 5th and 6th weeks of gestation, the spleen develops from the splenic anlage in the left dorsal mesogastrium, and the gonadal ridge is formed between the mesonephros and dorsal mesentery. During the 6th and 7th weeks of gestation, the dorsal mesogastrium rotates to the left, placing the splenic anlage into proximity with the left urogenital fold which contains the gonadal mesoderm. Such proximity remains until the descent of the gonads during the 8th week of gestation ,2,8. TheSGF can be classified into 2 types, based on its relationship with the native spleen. Continuous SGF is characterized by the presence of a cord of splenic or fibrous tissue connecting the spleen and gonad, and occasionally beads of splenic tissue could be found along the cord (splenic rosary bead), while in discontinuous SGF, there is no such connection. Rather, in discontinuous SGF, ectopic splenic tissue or accessory spleen is directly attached to the gonad without connecting to the native spleen ,34,35. 1SGF is often an incidental finding during exploration/surgery for scrotal swelling/mass, cryptorchidism, or inguinal hernia . In our We all know that SGF is a congenital disease, meaning that the benign tissue has existed and slowly developed along with the testis. Because it is a benign disease, preserving the testicle is always a top priority in treatment. However, according to a 1990 report, >37 % of SGF patients underwent an unnecessary orchiectomy . In our 4To the best of our knowledge, we present a case report of the continuous type of SGF, the first of its kind in Vietnam in the English literature. SGF is a rare benign birth defect that should be taken into account before performing orchiectomy, particularly when concurrent with cryptorchidism or inguinal hernia.Not applicable as it is a case report.This report involved no sources of funding for any of the authors.QN were the main doctors involved in conceiving the original idea; QN, DN, HN designed the study and gathered data. XB conceived the manuscript, edited it, performed the operation, and wrote the manuscript. All authors read and approved the final manuscript.Quang Nguyen.Written consent was obtained from the patient and the patient's father to publish this case report. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Herein we report the case of a patient with multiple glucagonomas that have been precisely described with endoscopic ultrasound. A 36‐year‐old woman was referred to our hospital for computed tomography investigation of multiple pancreatic masses. Physical examination was unremarkable; on contrast‐enhanced computed tomography, mass lesions were evident in the head, body, and tail of the pancreas. The mass in the pancreatic head was poorly demarcated and exhibited a faint contrast effect, the one in the pancreatic body was a cystic lesion, and the one in the pancreatic tail was hypervascular. Blood investigations showed that serum glucagon was abnormally high at 7670 pg/ml; glucose tolerance was not impaired. There was no family history that suggested multiple endocrine neoplasia type 1 or von Hippel‐Lindau disease. Endoscopic ultrasound revealed that there were additional masses, which were scattered isoechoic to hyperechoic lesions a few millimeters in size. Ultrasound‐guided fine needle biopsy of the lesion in the pancreatic tail resulted in a diagnosis of a neuroendocrine tumor. Based on these pathologic findings, we performed a total pancreatectomy. A large number of nodules with tumor cells were evident in all cut surfaces of the surgical specimen. Immunostaining was positive for chromogranin A and glucagon, and glucagonoma was therefore diagnosed. It is conceivable that attenuated glucagon action could have contributed to the development of the multiple glucagonomas. Pancreatic neuroendocrine tumors are a comparatively rare form of pancreatic tumors, accounting for less than 3% of all pancreatic tumors.A 36‐year‐old woman underwent a plain computed tomography (CT) scan to investigate pain in the left breast, which identified a pancreatic mass, and thus, she was referred to our hospital. The patient's medical history was unremarkable, and there was no family history of endocrine disease. On physical examination, her height was 160 cm and weight was 54.3 kg, vital signs were normal, and no abdominal symptoms or cutaneous lesions were present. The patient's blood count and biochemistry test results were within normal limits, and there were no elevated tumor markers. Endocrine hormone tests showed that serum glucagon was abnormally high at 7670 pg/ml. Furthermore, intact parathyroid hormone was only mildly elevated, and glucose tolerance was not impaired. Abdominal dynamic CT Figure . The solOn the basis of these imaging findings, multiple neuroendocrine tumors were suspected. Multiple endocrine neoplasia type 1 (MEN1) was ruled out through family history, absence of pituitary adenoma, and absence of hyperparathyroidism. The patient was admitted for an endoscopic ultrasound (EUS)‐guided fine‐needle aspiration. On EUS Figure , the lesN‐terminal‐recognizing and C‐terminal‐recognizing antibodies and has better specificity for glucagon than radioimmunoassay. Thus, hyperglucagonemia was confirmed with the sandwich ELISA method. From these results, we assumed that glucagon cell hyperplasia and neoplasia in this patient might be due to glucagon receptor abnormality. Previous studies reported that biallelic‐inactivating mutations in the glucagon receptor gene cause glucagon cell hyperplasia and neoplasia also termed Mahvash syndrome.Glucagonoma is an extremely rare tumor that accounts for approximately 1%–2% of neuroendocrine tumors.To date, there have been six reported cases of disease caused by biallelic mutations of the glucagon receptor gene,None.


Anal fistula is a common perianal disease, but primary complex anal fistulas with 7 external openings is extremely rare.We report a case of a 36-year-old man with a 10-year history of recurrent pus flow from paranal mass.Primary complex anal fistula.The patient underwent fistulotomy plus seton, which we successfully completed with the aid of 3-dimensional (3D) reconstruction model created from magnetic resonance imaging (MRI).The wound healed well and there was no recurrence 8 months after surgery.In the treatment of complex anal fistula, the combined application of 3D MRI model will be beneficial to obtain better surgical results. We report here a case of primary 7 external openings complex anal fistula with complete resection assisted by preoperative 3-dimensional (3D) magnetic resonance imaging (MRI) model. This case report presents a new treatment strategy for complex anal fistulas.Complex anal fistulas were defined as trans-sphincteric fistulas involving > 30% of the external anal sphincter, suprasphincteric fistulas, horseshoe fistulas, anterolateral trans-perineal complex fistulas in female patients, and fistulas with combined inflammatory bowel disease, radiation enteritis, malignant tumors, anal incompetence, chronic diarrhea.The patient, a 36-year-old male, was admitted to the hospital with a perianal mass with recurrent pus flow for more than 10 years. Over the past 10 years, the perianal masses occasionally increased in size and added multiple perianal masses with pain and ruptured pus, during which the patient received antiinflammatory treatment, but with poor results. Physical examination: the patient was placed in a lithotomy position, and a total of 7 masses were visible at 12, 1 to 3, and 5 o’clock sites, about 2 to 7 cm from the anus, with light pressure pain, ulcerated and flowing pus, and a cord-like mass extending into the corresponding anus .–11 However, later studies and their improved methods failed to replicate these results. Multicenter studies and Meta-analysis suggest that the success rate is <50%.,13 There are also surgeries for sphincter protection such as anorectal advancement flap, laser closure of anal fistula and so on.Currently, there are surgical methods for treating anal fistulas, including those that damage the sphincter such as: anal fistulotomy, fistulectomy, and seton, while the main surgical approaches to protect the function of the sphincter are: ligation of the intersphincteric fistula tract was first proposed by RojanasakulTherefore, in the treatment of complex anal fistula, it is necessary not only to choose the appropriate surgical method, but also to strengthen multidisciplinary communication and cooperation. The preoperative use of 3D reconstruction technology in imaging can guide surgeons to operate more precisely, reducing tissue damage and bleeding and shortening the operating time, thus increasing the cure rate of complex anal fistula and reducing recurrence. This primary fistula has a long course, evolving over a period of 10 years into a complex fistula with 7 external openings, which reminds us that fistulas should be treated early to avoid aggravation of the condition.Data curation: Yonggang Qiu.Visualization: Yonggang Qiu.Writing – original draft: Yongping Hong.Writing – review & editing: Guofeng Li.


Primary hyperparathyroidism (PHPT) is the unregulated overproduction of parathyroid hormone (PTH), resulting in abnormal calcium homeostasis. PHPT is most commonly caused by a single adenoma of the parathyroid gland, which can have an intrathyroid location in rare cases. The measurement of intact PTH in the washout fluid obtained by ultrasound (US)-guided fineneedle aspiration (FNA) can be useful in clarifying the aetiology of these lesions. This study presented a 48-year-old man with a background history of symptomatic renal stone disease who was diagnosed with PHPT and referred to our Endocrinology department. A neck US revealed a thyroid nodule with a size of 21 mm in the right lobe. The patient underwent US-guided FNA of the lesion. The measurement of PTH in the washout fluid was significantly elevated. Following the procedure, he reported neck pain and noticed distal paraesthesias in the upper limbs. Blood test results showed significant hypocalcaemia and supplementation with calcium and calcitriol was started. The patient was closely monitored. Recurrence of hypercalcaemia was later observed, and the patient was submitted to surgery. We present a case of FNAinduced transitory remission of PHPT in a patient with an intrathyroid parathyroid adenoma. We conjecture that intra-nodular haemorrhage might have occurred, which temporarily affected the viability of the autonomous parathyroid tissue. A few similar cases of spontaneous or induced remission of PHPT after FNA have been previously described in the literature. This remission can be transitory or permanent, depending on the degree of cellular damage thus follow-up of these patients is recommended. Hyperfunctioning parathyroid lesions are susceptible to fine-needle aspiration (FNA)-induced damage.FNA biopsy is not generally recommended for the evaluation of primary hyperparathyroidism (PHPT), although it might be useful in selected cases, particularly by measuring parathyroid hormone in the washout.Considering the high recurrence rate, close follow-up of patients with FNA-induced remission of PHPT is recommended.99mTc-sestamibi scintigraphy and neck ultrasound (US). US is largely used in these patients as it is widely available, does not involve ionizing radiation and has a high sensitivity to detect these lesions (Primary hyperparathyroidism (PHPT) is a common endocrine disorder characterized by hypercalcaemia due to an unregulated overproduction of parathyroid hormone (PTH). PHPT is most commonly caused by a single adenoma of a parathyroid gland but can be caused by PHPT-related multiglandular disease in 10–15% of the cases. Parathyroid carcinoma is rare and occurs in less than 1% of the cases  and PTH 462.1 pg/mL  and referred to our Endocrinology department. He presented a background history of symptomatic renal stone disease and had already been submitted to lithotripsy.A neck US was performed, and the only abnormal finding was a 21 mm predominantly cystic thyroid nodule in the right lobe A and B. 99mTc-sestamibi scan was performed and did not reveal any abnormalities suggestive of parathyroid disease  and reduction of PTH levels to 124 pg/mL. The patient reported symptomatic improvement after starting therapy with calcium plus vitamin D. Concurrently, a  disease . SequentThe patient was submitted to right thyroid lobectomy. During surgery, serum PTH decreased from 178.8 to 15 pg/mL within 10 min of the excision. The postoperative course was uneventful, with the exception of transient hypocalcaemia.Histopathology examination confirmed the presence of an intrathyroid parathyroid adenoma. The lesion had areas of fibrosis and hemosiderin deposition consistent with prior puncture .Figure Soon after surgery, calcium and PTH levels were within the normal reference range and have remained so ever since (last follow-up 17 months after surgery). 99mTc-sestamibi scan was performed during remission of PHPT, which may explain the negative result.We present a case of FNA-induced transitory remission of PHPT in a patient with an intrathyroid hyperfunctioning parathyroid adenoma. Serum PTH and calcium dropped from 462.1 to 124 pg/mL and 12.0 to 8.1 mg/dL, respectively, following FNA. These unexpected changes along with neck pain suggest that FNA-induced haemorrhage led to remission of PHPT, which temporarily has affected the viability of the autonomous parathyroid tissue. Remarkably, the PTH level gradually began to rise 1 month after the procedure, demonstrating the damage was only temporary. In our case, a Spontaneous remission of PHPT due to nontraumatic necrosis, haemorrhage and infarction of a parathyroid adenoma is extremely uncommon, but it is a previously well-documented event, usually denominated as ‘parathyroid autoinfarction’, ‘autoparathyroidectomy’ or ‘parathyroid apoplexy’. This rare incident may range in presentation from asymptomatic to life threatening, with signs and symptoms of massive cervical or mediastinal haemorrhage, a condition requiring emergency neck exploration surgery. Most of the reported cases presented with acute hypocalcaemia, resulting from the adenoma’s necrosis and ineffectiveness of the remaining parathyroid glands to produce PTH, followed by a period of normocalcaemia at first and finally recurrence of the disease. Surgical treatment was performed in most cases, with only a few cases reported in which regular follow-up was initially chosen. This indicates that the apparent cure that follows necrosis of a parathyroid adenoma is potentially temporary, possibly explained by the presence of non-ischaemic adenomatous tissue having a potential to grow, resulting in the recurrence of hypercalcaemia at some stage. Therefore, long-term clinical and biochemical surveillance is advised (In our case, remission of PHPT was caused by FNA of the parathyroid adenoma. This phenomenon is exceptional, and to the best of our knowledge, only six cases have been published so far .This remission can be transitory or permanent, depending on the degree of cellular damage. The patients reported by FNA is usually not recommended for parathyroid suspected tumours due to the chances of serious complications, such as massive haematoma, parathyromatosis and misdiagnosis as malignancy during pathological diagnosis. However, when the localization is unusual or parathyroid adenoma is mistaken for a thyroid nodule, FNA may be indicated or performed erroneously. Although cytomorphology alone may help distinguishing between parathyroid and thyroid lesions, it has remained a great challenge due to overlapping features (In conclusion, our case, together with a literature review, suggests that hyperfunctioning parathyroid lesions are susceptible to FNA-induced damage. This procedure is not generally recommended for the evaluation of PHPT, although it might be useful in selected cases. Close follow-up of patients with FNA-induced remission of PHPT is recommended, considering the high recurrence rate.The authors declare that there is no conflict of interest that could be perceived as prejudicing the impartiality of this case report.This work did not receive any specific grant from any funding agency in the public, commercial or not-for-profit sector.Written informed consent for publication of their clinical details and clinical images was obtained from the patient.A R E drafted the manuscript. J C and F J C R were involved in critical revision of all drafts of the manuscript. J C, B M, R C M, J S, T M and J G were involved in patient care. All authors have approved the final version of the manuscript.


Osteosarcoma of the foot is exceedingly uncommon, and as a result, very little is known about patient and tumor characteristics. In addition, the prognosis may be grim due to delayed presentation and misdiagnosis. A delayed diagnosis of osteosarcoma, regardless of location, may not only reduce long-term survival but also modify the treatment plan, resulting in less favorable functional and cosmetic outcomes. Here we report the diagnostic and therapeutic challenges associated with chondroblastic osteosarcoma involving the metatarsal bone of the foot in a 47-year-old woman treated with wide local excision with right second metatarsectomy and non-vascularized fibular graft reconstruction along with adjuvant chemotherapy. Foot and small bone osteosarcomas are a rare and distinct clinical entity. The incidence of foot osteosarcomas is estimated to range between 0.2% and 2% . ApproxiA 47-year-old woman presented to the surgical oncology outpatient department with a painful, progressive swelling on the dorsum of the right foot of two months duration. She had no history of trauma or fever. The patient had a 5x3 cm bony hard mass arising from the second metatarsal bone of the right foot on the dorsal aspect. There was no evidence of popliteal or inguinal lymphadenopathy and distal motor or sensory deficits. All distal pulses were equally felt. She presented us with a fine-needle aspiration cytology (FNAC) report from the soft tissue component of the metatarsal swelling, which showed features of a giant cell tumor (GCT). The plain radiograph revealed an expansive cortical diaphyseal lesion in the right second metatarsal with punctate calcification and a periosteal reaction that was suggestive of osteosarcoma Figure .The chest radiograph of this patient was normal. Due to the patient's financial constraints, an MRI of the foot could not be carried out, and we proceeded with the surgery. A wide local excision with the right second metatarsectomy was performed (R0 en-bloc resection). The foot was reconstructed using a 6 cm non-vascularized fibular graft using k-wire Figures , 3.The postoperative period was uneventful. On the fifth postoperative day, she was able to commence partial weight bearing, and on the 14th postoperative day, her sutures were removed. The k-wire was removed six weeks after surgery  protocol. As the resection margins were tumor-free, no adjuvant radiation therapy was necessary.We reported the case of a 47-year-old woman with a painful swelling on the dorsum of her right foot, which was a bony, hard mass from the second metatarsal bone. FNAC suggested a GCT, although conventional radiography suggested osteosarcoma. An MRI scan was unaffordable. Wide local excision with right second metatarsectomy and non-vascularized fibular graft reconstruction were performed. Histopathology indicated chondroblastic osteosarcoma with clear margins after a smooth recovery. The patient received adjuvant chemotherapy, and no radiation therapy was required due to tumor-free resection margins.The clinical presentation of osteosarcoma of the foot bones is non-specific. Schuster AJ et al.  analyzedRadiographically, osteosarcoma can mimic benign conditions such as aneurysmal bone cysts and chondroblastomas . Plain rSeveral differential diagnoses, including benign tumors, must be considered when diagnosing and treating such patients. GCTs and chondromyxoid fibroma are the two most prevalent benign tumors affecting the metatarsal . GCTs caOur patient underwent a wide local excision in the form of a second metatarsectomy, and reconstruction was done using a non-vascularized fibular graft. This choice was made due to its easier operative technique, shorter surgical time, and proven high union rates for patients up to 12 cm . There iBased on the final histopathological diagnosis of chondroblastic osteosarcoma, adjuvant chemotherapy was recommended for our patient. In accordance with the established osteosarcoma treatment protocol, the administration of neoadjuvant and adjuvant chemotherapy has become the standard strategy for improving overall survival rates and minimizing the risk of metastasis . Due to It is crucial to acknowledge the limitations of our study, including the lack of MRI due to financial constraints. The MRI provides vital information about the tumor's extent and facilitates surgical planning. Despite this limitation, we were able to achieve clear surgical margins with wide local excision. Furthermore, the follow-up period in our case is relatively short, and long-term outcomes are yet to be determined.In conclusion, this case report illustrates the challenges associated with diagnosing and treating chondroblastic osteosarcoma involving the metatarsal bone. Comparisons with similar studies highlight the significance of an accurate histopathological diagnosis, wide local excision with reconstruction, and adjuvant chemotherapy in attaining favorable outcomes. To optimize treatment for metatarsal osteosarcoma, additional research and larger-scale studies need to be conducted to expand our understanding of this rare entity and optimize treatment options.


The extended transforaminal endoscopic approach allows visualization and manipulation of the third ventricle posterior structures in a safe and comfortable manner. The medial subchoroidal approach has been described as a feasible alternative to the classical transchoroidal approach.In this video, the authors present the case of a 14-year-old male with a history of neurofibromatosis type 1 who was referred to our department after presenting with headaches and diplopia for 2 weeks. Suspecting an aqueduct tumor, the authors performed an endoscopic surgical procedure through a single approach with third cisternostomy and resection of the tumor that produced the stenosis.https://stream.cadmore.media/r10.3171/2023.1.FOCVID22155The video can be found here:  We present the feasibility of an extended transforaminal approach  for resection of a benign aqueductal tumor in a patient with type 1 neurofibromatosis. No relevant financial disclosures are declared.This is a 14-year-old male with a history of type 1 neurofibromatosis followed by multiple neuromas in the lumbar plexus. He was referred to our department after presenting with headaches and diplopia for 2 weeks. His physical exam was remarkable for a right cranial nerve VI palsy with no other focal symptoms. Funduscopic examination revealed incipient bilateral papilledema. An emergent MRI was performed showing a triventricular hydrocephalus with a suspected lesion in the upper part of the aqueduct of Sylvius.The T2-weighted images show dilatation of the ventricular system with associated transependymal edema and signs of endocranial hypertension. The gadolinium-enhanced sagittal T1 sequences show no significant enhancements without clear pathology. Nevertheless, in sagittal CISS sequences, we can observe a normal-sized fourth ventricle with the suspicion of a lesion in the most cranial part of the aqueduct marked by the red arrow.1 Because we want to add a ventriculostomy during the surgery, we plan a trajectory that is the bisector of both trajectories to both desired targets with a single burr hole in the precoronal position. A rigid endoscope was used with a 6° view angle .The surgical planning. We use general anesthesia with the patient in a supine position. The head is slightly elevated (about 20°) with a right frontal arciform incision. No Mayfield clamp is used, and electromagnetic navigation is preferred . For the extended transforaminal approach, it is important to measure the size of the foramen of Monro because it is a marker of the difficulty we will encounter during the procedure.4 The measures of foramen of Monro are shown in this image. In this case, the trephine was made 1.7 cm anterior to the coronal suture and 2.8 cm from the midline as shown in this 3D CT reconstruction.The choice of trajectory is shown in this picture. The two ideal trajectories would require two different burr holes, but the extended transforaminal approach allows us to simplify the surgery by performing only one burr hole.Once we enter the ventricle, we perform a careful inspection of the structures we encounter. We can see a dilated foramen of Monro showing the third ventricle floor structures. We identify structures that let us confirm that we are in the right lateral ventricle, such as the septal vein, the choroid plexus, and the thalamostriate vein.5In this case, the confluence of the septal and the thalamostriate vein allows us to extend the foramen lateral to the choroid plexus and medial to the thalamostriate vein, in a medial subchoroid fashion, thus reaching the posterior third of the third ventricle.With gentle and delicate maneuvers, we can dissect the tenia choroidea in a safe manner, avoiding large tractions on the vascular nerve structures and reaching the posterior part of the third ventricle. A blunt dissection is performed by the endoscope itself without no need to coagulate veins. At this point, we can identify the aqueduct of Sylvius and a tumor in the upper part that clearly obstructs the cerebrospinal fluid circulation.Surgical excision of the lesion is performed with standard tools.Once the lesion is resected, patency of the aqueduct is observed with free circulation of cerebrospinal fluid through it. No complications secondary to resection were observed.At this point, we decided to add a safety ventriculostomy by guiding the endoscope to the anterior part of the floor of the third ventricle. Mammillary bodies and premammillary membrane are identified. With the bipolar tool, we perforate the membrane and then use a 4-Fr Fogarty to dilate the perforation. Once completed, the endoscope is introduced to confirm the correct perforation of the Liliequist membrane. The basilar artery is identified with no significant surgical complications.6 The medial subchoroid approach is shown without damage to the venous structures. Intraoperative reduction of the ventricular size is observed.When the endoscope is removed, the absence of lesions on the fornix is confirmed.Clinical outcome and follow-up. The postoperative showed no complications, and the patient was discharged 72 hours after surgery. Evident improvement of the headache was observed and the sixth nerve palsy resolved 1 month after surgery. Follow-ups at 6, 12, and 24 months confirmed excellent radiological and clinical evolution with no signs of tumor recurrence.After 2 years, the T2-weighted imaging sequence shows complete resolution of the hydrocephalus. In the sagittal plane, it is possible to confirm the patency of the ventriculostomy and the associated flow artifact that can also be seen in the aqueduct. Thank you.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: Narro-Donate. Editing and drafting the video and abstract: Narro-Donate. Critically revising the work: Urreta-Juarez, Castelló-Ruiz, Masegosa-González. Reviewed submitted version of the work: Narro-Donate, Guil-Ibañez, Castelló-Ruiz, García-Pérez, Urreta-Juarez. Approved the final version of the work on behalf of all authors: Narro-Donate. Supervision: Masegosa-González. Video narration: Narro-Donate.The necessary patient informed consent was obtained in this study.


Isolated neglected subcutaneous rupture of the flexor digitorum superficialis (FDS) in zone III of the hand is a rare injury that is distinct from subcutaneous rupture of the deep flexor tendons in the fingers. While a few cases have been reported in the literature, the pathophysiology of this injury remains poorly understood.In this article, we present a case study of a climber who experienced an isolated subcutaneous rupture of the FDS following a sports accident. The patient's diagnosis was initially delayed due to an unclear clinical presentation. However, surgical intervention was successfully performed, resulting in positive clinical outcomes at the one-month follow-up. This case highlights the importance of considering isolated subcutaneous rupture of the FDS as a potential injury in climbers and other individuals who engage in high-impact sports. Although isolated neglected subcutaneous rupture of the flexor digitorum superficialis (FDS) is often considered a rare injury, it should not be disregarded . While aIn this article, we present a case study of a climber who experienced an isolated subcutaneous rupture of the FDS in zone III of the hand. The patient was treated surgically and had a favorable outcome.We present the case of a 38-year-old right hand dominant climber with no comorbidities who visited the emergency department after experiencing a cracking sensation in the third finger during a training session. Standard radiography did not reveal any abnormalities Figure . A preliAfter three weeks, the patient had no improvement and continued to experience pain and limited range of motion in the affected finger. An ultrasound was performed, which did not reveal any tendon rupture. Conservative treatment was continued with physiotherapy.Two months later, the patient presented to our hospital with persistent discomfort during the flexion of the third finger and a nodule in the third zone of the flexors in the left hand. Clinical examination showed complete but difficult flexion of the finger. A comparative hand examination revealed a deficit in flexion of the proximal interphalangeal joint compared to the healthy side.An MRI confirmed an isolated rupture of the third tendon of the FDS in the left hand, which had retracted to zone III. The flexor digitorum profundus tendon was found to be intact Figure , and surUnder locoregional anesthesia, a Z-shaped incision was made over the nodule. The stump of the ruptured tendon was found in zone III , and it was resected due to its poor quality and retraction. The flexor digitorum profundus was embedded in fibrotic scar tissue, so tenolysis was performed and pulleys were intact Figure .The postoperative course was simple, no immobilization was done, a self-rehabilitation was started on day 2 post-operation after the subsidence of the pain.The patient was followed up for two years. No skin complications were reported Figure , and theNon-pathological subcutaneous rupture of the FDS of the fingers is a rare diagnosis in current hand surgery practice, and few cases have been published in the literature. In 2007, Bois et al.  reviewedSeveral hypotheses have been proposed to explain the rarity of non-pathological ruptures of the FDS at the fingers. One such hypothesis is that the footprint of the FDP on the distal phalanx is narrower than that of the FDS on the middle phalanx, thus making the FDP more important in finger flexion and more susceptible to injury . Folmar The clinical presentation is almost always the same: deformation of the proximal inter phalangeal joint in flexion, persistent pain in the hand and reduced range of motion of the proximal inter phalangeal joint . In mostThe role of standard radiography in the diagnosis of non-pathological ruptures of FDS of the fingers is to detect a bone avulsion at the FDS insertion and assess the condition of the interphalangeal joint. Ultrasound is useful for confirming the diagnosis, quantifying retraction, and identifying the location of the rupture, which is often at the level of the A1 pulley or in Camper's Chiasm. It can also be used to assess for associated pulley or FDP lesions. While MRI is not always necessary, a well-performed ultrasound by a trained radiologist can provide the information needed for surgical planning.An anatomical classification of this lesion was proposed by Alsaadi et al.  based onThe surgical technique proposed by Stern et al.  involvesCaso et al.  reportedOur case is an isolated non-pathological FDS tear with delayed diagnosed. We believe this lesion is underestimated, and its incidence is greater than believed, so we insist that each patient must undergo a comparative clinical examination of both hands irrespective of their pathology. The diagnosis is generally clinical, and the treatment is classical, including the release and excision of the cicatricial fibrosis. No postoperative immobilization is necessary thus a self-rehabilitation of the fingers must begin immediately.Although isolated neglected subcutaneous rupture of the FDS is considered rare, we believe that its incidence may be greater than believed. We believe that a careful, bilateral, and comparative clinical examination in the face of even minimal hand trauma and prompt surgery can prevent prolonged and potentially irreversible disability.


Venous malformations are the most common type of congenital vascular lesions resulting from abnormal embryonic development of vessels. Typical venous malformations are easily diagnosed by skin color changes, focal edema, or pain as they are mostly present in the skin and subcutaneous tissue. Venous malformations in the skeletal muscles, however, have the potential to be missed because their involved sites are invisible. We describe a 15-year-old patient with extensive intramuscular venous malformations in the lower extremity with special emphasis on diagnosis and treatment. Vascular anomalies in children are of two varieties: proliferative or vascular tumors (eg. hemangiomas) and vascular malformations . VasculaHerewith, we report a case of a deep intramuscular VM (IMVM) that went undiagnosed in a paediatric patient until he was older but was successfully managed with surgical intervention and rehabilitation.A 15-year-old male was evaluated for left knee pain and difficulty walking for the past six years. He did not have any history of previous trauma or other pertinent past medical or surgical illnesses. Physical examination demonstrated, a severe flexion deformity, with a range of motion of −90° - 135° of flexion in the left knee Figure . WastingAfter discussing treatment options with the family, intervention radiology, cardio-thoracic and vascular surgery teams, surgical resection of the venous malformations was performed. The surgery was performed under general anesthesia in prone position on a regular operating table. The incision was placed on the posterior aspect of the thigh, from mid-thigh extending distally. After blunt dissection of the layers, care was taken to identify and preserve the sciatic nerve. Sparing the tendons, necrosed parts of hamstring muscles and vastus lateralis were identified and excised along with the supplying vessels. Resected specimen (of fibromuscular soft tissue with attached subcutaneous adipose tissue measuring 20x12x6cm) showed skeletal muscle fibers, adipose tissue, numerous irregularly elongated, dilated, and thick-walled capillaries with chronic inflammation suggestive of arterio-venous malformation on histopathology Figure . This waAt the end of three months, our patient was able to achieve complete extension Figure . His casIntramuscular venous malformations can result in severe co-morbidities if left undiagnosed for a long time. The diagnosis of this condition, however, is very challenging due to their young age at presentation and due to the deep location of the lesions . Hence, Though intramuscular VMs may be present at birth and are believed to be benign, they have the potential to grow in children during periods of their growth . They arIMVMs can be difficult to diagnose and treat, due to delayed presentation, deep location and vague symptoms. A thorough physical examination, followed by an appropriate investigative workup is necessary for timely diagnosis. Most IMVMs are asymptomatic and some may present with pain at the site of the lesion. In that case, traumatic injury, myositis and fasciitis must be ruled out. It is also crucial to be able to differentiate a vascular malformation from a malignancy. In a lesion that presents as a mass in an extremity, an MRI followed by a biopsy of the lesion should be done if the diagnosis is suspicious. On diagnosis of an IMVM, a treatment plan is to be formulated, keeping in mind the patient's age, presenting symptoms, location of the lesion, and extent of the deformity. In our patient, a surgical excision with deformity correction followed by regular follow-up resulted in full recovery. Therefore, though our patient’s condition went undiagnosed for a long time due to the lack of awareness and his limited resources, when presented with opportunities to avail the necessary expertise, a prompt diagnosis and successful intervention were done to help with a complete recovery from his presentation of a large intramuscular venous malformation.


Abdominal pain in pregnant individuals presents diagnostic challenges, especially when appendicitis is suspected. We report a rare case of a 26-year-old pregnant female with recurrent right lower quadrant (RLQ) abdominal pain initially misdiagnosed as a urinary tract infection. Diagnostic uncertainty led to a magnetic resonance imaging (MRI) scan, which revealed a right adnexal cystic structure and a thickened tubular structure adjacent to the cecal pole, raising concerns of complicated appendicitis. Subsequent diagnostic laparoscopy revealed a right-sided fallopian tube paratubal cyst with 360-degree torsion and associated fallopian tube torsion without the involvement of the ovary. The cyst was successfully excised, and the patient subsequently delivered a healthy baby via emergency lower section caesarean section.Abdominal pain during pregnancy has various causes. Diagnosing appendicitis during pregnancy is challenging due to anatomical and physiological changes. Ultrasound (US) is commonly used but has limited accuracy. Computed tomography (CT) is avoided due to radiation risks, while MRI is increasingly used and shows high diagnostic accuracy or aids in alternative diagnoses. Regardless of the diagnosis, the prompt recognition of intraabdominal pathology is crucial to prevent fetal morbidity.This case highlights the challenges in the accurate diagnosis of abdominal pain during pregnancy and emphasizes the importance of considering alternative pathologies to prevent delays in treatment and complications. Clinicians should consider diagnostic laparoscopy for pregnant patients with equivocal investigations and lower abdominal pain. The differential diagnosis may include both common and rare causes such as concomitant paratubal cyst and isolated fallopian tube torsion (IFTT), emphasizing a high index of suspicion and collaboration with obstetric colleagues to ensure optimal care. Abdominal pain during pregnancy presents a complex diagnostic challenge. Similar to non-pregnant females, abdominal pain in pregnant individuals can arise from various organs and pathological processes. However, one crucial diagnosis that requires diligent evaluation is appendicitis. Healthcare professionals frequently seek a general surgery consultation when confronted with abdominal pain during pregnancy, primarily to exclude the possibility of appendicitis. Recent reports indicate that appendicitis occurs in approximately one in 1500 pregnancies . Obstetr6/L; erythrocytes, <10 × 106/L; epithelial cells, >50 × 106/L) and abdominal pain. She was commenced on oral cefalexin and discharged home. Two days later, she re-presented with an episode of severe RLQ abdominal pain with associated nausea and vomiting. She was given intravenous ceftriaxone and, due to carer responsibilities, discharged home. She once again re-presented on day 5 of the initial onset of symptoms. On arrival, she was found to have tachycardia of 120 beats/minute and otherwise normal observations. Her abdomen demonstrated laparoscopic scars, with a gravid uterus palpable above the umbilicus. She demonstrated tenderness and percussion tenderness in the RLQ of the abdomen, particularly over a palpable mass. She underwent an ultrasound (US) scan of her pelvis, which reported a right adnexal cystic structure measuring 55 × 52 × 37 mm, presumed to be a simple ovarian cyst. Despite continued antibiotics and opioid analgesia, blood investigations demonstrated a C-reactive protein (CRP) of 114 g/dL with a normal white cell count. A general surgery consult was arranged as complicated appendicitis was proposed as a differential diagnosis by the treating gynecology team. Her persistent abdominal pain and clinical suspicion necessitated a magnetic resonance imaging (MRI) scan of her abdomen  abdominal pain for six days. Her pregnancy was also complicated by contracting cytomegalovirus, with close monitoring enacted to ensure appropriate fetal growth. The patient presented to the pregnancy assessment center six days prior to the presentation and was diagnosed with a urinary tract infection based on a contaminated urine sample  scan is the initial imaging modality employed for assessing acute appendicitis in pregnant females due to its accessibility and cost-effectiveness. However, its sensitivity and specificity in this context are limited. Computed tomography (CT) scans are generally avoided due to potential harm from ionizing radiation exposure to the fetus. Conversely, the utilization of magnetic resonance imaging (MRI) to diagnose appendicitis during pregnancy has increased in recent times. MRI has been reported to exhibit a sensitivity ranging from 90% to 100% and a specificity ranging from 87% to 98% for diagnosing acute appendicitis in pregnancy and thus is an important investigation . While oAbdominal pain during pregnancy can have various causes, some of which can also result in fetal morbidity. Paratubal cysts, which are typically benign fluid-filled sacs lined by ciliated cuboidal cells originating from Mullerian or Wolffian remnants , are oftIn summary, general surgeons should have a low threshold to consider diagnostic laparoscopy in patients with equivocal blood and imaging investigations who have lower abdominal pain during pregnancy. The differential diagnosis is wide and varied, and many pathologies may masquerade as appendicitis. While rare, paratubal cysts as a cause of IFTT should be considered in the differential diagnosis of abdominal pain in pregnancy, especially when a presumed ovarian cyst is observed on imaging. A high index of suspicion and collaboration with obstetric colleagues is necessary to preserve future fertility and avoid morbidity of the pregnant mother and fetus.


A 41-year-old, New Zealand European male, was seen in the acute eye clinic as an in-patient from the trauma ward with multiple comorbidities including an orbital fracture from injuries sustained in a road traffic accident. It is not uncommon in emergency settings for emergency physicians to review polytrauma patients prior to Ophthalmologists, with Computerized Tomography being the imaging modality of choice. A hyper-dense lesion within the right globe was noted by radiology at the time and concerns were raised about the possibility of a retained intraocular foreign body. Upon Ophthalmic examination, a clinical diagnosis of sclerochoroidal calcification was made. This case highlights a rare case of sclerochoroidal calcification manifesting as a hyperdense lesion on computerized tomography masquerading as an intraocular foreign body. Globally it is estimated that 55 million eye injuries occur annually Radiographic evaluation in the assessment of intraocular foreign bodies is essential, as often patients may present with polytrauma, and history or physical examination can often be limited by the severity of the injuries. Magnetic resonance imaging of any metallic foreign body is contraindicated due to the magnetic field generated causing any metallic material to become dislodged, damaging ocular structures along its trajectory This case highlights sclerochoroidal calcification, an idiopathic and usually benign ocular condition that masqueraded as an intraocular foreign body on CT imaging of a patient presenting with polytrauma.A 41-year-old, New Zealand European male, was seen in the acute eye clinic as an in-patient from the trauma ward with multiple comorbidities sustained from a car vs bicycle collision. the patient ejected off their bicycle and sustained a multitude of injuries relating to multiorgan perforations, lacerations, and fractures. Most notably, a right tripod facial fracture was detected with concerns raised as to the integrity of the globe and its structures.Once the patient was stabilized, follow up examinations for the tripod fracture were arranged by the Maxillofacial department with Ophthalmology input requested to investigate the possibility of a retained IOFB, given the hyperattenuating lesion noted in the right eye on CT imaging .Fig. 1 imaging through the right retinal lesion was obtained  which shA diagnosis of a right unilateral scleral choroidal calcification at the site of the superior oblique insertion was made with no globe injury related to the trauma. At maxillo-facial review, a decision was made not to repair the orbital fracture given the absence of diplopia and enophthalmos. The patient was discharged back to the care of their optometrist for regular review included dilated fundoscopy and EDI-OCT scans of the sclerochoroidal lesion.This case highlights 2 main points: Ocular injuries and to a lesser extent retained intraocular foreign bodies, are extremely common in tripod facial fractures, requiring acute ophthalmology collaboration. Secondly, increasing the recognition of sclerochoroidal calcification as a masquerading condition to retained intraocular foreign body amongst other differential diagnoses.Ocular injuries are common amongst facial fractures, highlighting the need for acute ophthalmology input. Al-Qurainy and colleagues  demonstrCT is the most practical step in assessing trauma cases as it is readily available and can detect not only osseous damage but a wide range of metallic intraocular foreign bodies safely at small limits of detection . Most meThe challenge arises when other conditions, such as sclerochoroidal calcification, masquerade as retained intraocular foreign bodies due to similar hyper dense appearances on CT imaging. Sclerochoroidal calcification is an idiopathic, usually benign, yet uncommon condition characterized by yellow-whitish subretinal lesions classically located in the superior temporal midperiphery of the fundus beyond the vascular arcades . DespiteIn some rare cases, there may be systemic conditions associated with metastatic sclerochoroidal calcification usually involving abnormal calcium phosphorus metabolism or renal tubular hypokalemia metabolic alkalosis syndromes . PatientThis case is a rare example of a singular unilateral lesion found in a uniquely young individual, under exceptional circumstances. Due to the complicated nature of this polytrauma scenario, specifically affecting the periorbital region, an initial diagnosis of intraocular foreign body was made based on CT findings. This case not only highlights this poorly understood entity, but it also demonstrates the significant power of interspecialty collaboration alongside multimodal imaging techniques on masquerading conditions.In summary, sclerochoroidal calcification is a rare and usually benign condition which may masquerade as a retained intraocular foreign body as in the case of this patient. As CT is usually the first imaging modality used in trauma cases, radiologists are among the first to assess scans and as such hyper dense lesions within the globe are more commonly thought to be as a result of retained intraocular foreign bodies, therefore adding complexity in differential diagnoses. Furthermore, this case highlights the benefits of multimodal imaging and likewise demonstrates a typically rare ocular condition of sclerochoroidal calcification.Written informed consent was obtained from the patient for the publication of this case report.


Gordonia spp. are gram-positive bacteria that have previously been reported to cause mycetoma. Here, we report a case of Gordonia soli  as the etiological agent of actinomycetoma in a 64-year-old patient. After a literature search in the Cochrane Library, LILACS, SciELO, MEDLINE, PubMed, and PubMed Central databases, we concluded that this is the first case report of mycetoma caused by Gordonia soli. The current case highlights the importance of microbiological diagnosis of mycetoma and the challenges in its management.Mycetoma is a neglected tropical disease caused by fungi (eumycetoma) or bacteria (actinomycetoma), with high morbidity.  The route of infections is usually the transcutaneous inoculation of sapronotic organisms, including several species of bacteria and fungi. The etiologic agents of mycetoma usually are traumatically inplanted trhough splinters, thorns an plant debris.The most commonly involved anatomical regions are the legs and feet, although the shoulders and upper back may also be affected when carrying wood logs or lying down in direct contact with the soil in endemic regions,Most mycetoma cases have been reported in Mexico, India, and Sudan,Nocardia brasiliensis, Streptomyces somaliensis and Actinomadura madurae,The presumptive diagnosis of the microbiologic etiology of both actinomycetoma and eumycetoma can be made based on the morphological features of direct grain microscopic examinationGordonia soli, initially identified as Nocardia spp. Additionally, a literature review was performed using the Cochrane Library, LILACS, SciELO, MEDLINE, PubMed, and PMC  databases.Here, we present a case of mycetoma caused by Nocardia spp. based on clinical and histopathological findings.A 64-year-old man with no comorbidities presented with painful nodular lesions on his left foot following a traumatic injury. On examination, there were enlarging nodules that progressed to purulent lesions with sanguinolent discharge, draining fistulas, and elimination of grains. A clinical diagnosis of mycetoma was made and a biopsy was performed. The histopathological evaluation revealed granulomas and fibrosis. It was also remarkable for the presence of grains and filamentous structures compatible with an actinomycetoma (Gordonia soli was recovered by culture and identified using 16S rRNA gene sequencingThe patient was treated with trimethoprim-sulfamethoxazole (TMP-SMX) for eight years, without complete resolution of symptoms, as he had persistent painful nodular draining lesions . A new cGordonia identified by MALDI-TOF MS (score 1.62) (Bruker MALDI Biotyper), which was confirmed as Gordonia soli by 16S rRNA gene sequencing, indicating persistence of infection. Magnetic resonance imaging (MRI) of the foot revealed signs suggestive of inflammation in the subcutaneous tissue; midtarsal and tarsometatarsal joints; and the calcaneum, midfoot, and metatarsal bones, suggesting osteomyelitis. The “dot-in-circle” sign was present, which is suggestive of mycetoma  and species levels (>1.7) was proposed,,-Gordonia are present in the soil and can be transferred through aerosols from environmental sources to the respiratory tract, thereby causing pulmonary infections, mainly in immunocompromised hosts. They can also cause bacteremia and spread to the central nervous system, cardiac tissues, and joints. G. sputi, G. bronchialis, and G. terrae are the most frequently identified species in clinical samplesMembers of the genus ,Gordonia can be performed using broth microdilutions. Based on isolates reported in literature, it is most predictably susceptible to imipenem, ciprofloxacin, amikacin, linezolid, gatifloxacin, gentamicin and vancomycinin vitro and surgical therapy was considered. However, this was not performed after a shared decision-making approach with the patient, as it was predicted to be mutilating.Actinomycetoma is usually treated with long-term anti-infective medications. Recurrence is common and some patients require combined surgical management to achieve clinical successGordonia have been reported: two caused by Gordonia terrae and one by Gordonia weatfalia,. To the best of our knowledge, this is the first reported case of actinomycetoma caused by Gordonia soli.Three cases of actinomycetoma caused by


Dear Editor,Blue rubber bleb nevus syndrome (BRBNS) is characterized by multiple venous malformations that often affect the skin and gastrointestinal tract, in some cases causing intestinal bleeding and iron-deficiency anemia.An 11-year-old female had a history of bluish lesions on the right earlobe and left thigh since birth. The parents reported progressive increase in lesion size over the years and the appearance of new similar lesions. The clinical examination showed a violaceous lesion on the left thigh with slight swelling on palpation, measuring 4×1 cm, a violaceous papule on the right earlobe measuring 1.5×1 cm, discrete violaceous spots on the lumbar region and a small violaceous spot on the right shoulder, measuring 0.5 cm . The patA 13-year-old healthy female was referred to the Dermatology Service for evaluation of a multifocal vascular lesion in the cervical region, present since birth. She reported the recent appearance of new lesions on her face and right thigh . The ComBRBNS, also known as Bean syndrome, is a rare condition, in which multiple vascular malformations usually arise in the skin and digestive tract, and which can also affect other organs such as the heart, liver, spleen, eyes, central nervous system, lung, bladder, thyroid and parotids. The lesions appear more frequently in the skin and small intestine and may lead to digestive bleeding and iron deficiency anemia. The lesions may be present at birth but tend to increase in size and number throughout life.Both primary lesions are described as bluish papules with vascular appearance and varying sizes. Both types of lesions tend to be painless, but painful lesions are not uncommon. Glomangiomas can be painful during pregnancy and menstrual periods.Autosomal dominant inheritance has also been demonstrated in both diseases.9On histopathology, dilated vascular spaces and the presence of glomus cells lining the endothelium are unique to glomangiomas, together with positive immunohistochemistry for SMA (marker of actin-related protein present in smooth muscle), caldesmon  and myosin (marker of protein present in muscles), which confirms the muscular nature of these cells. These immunohistochemical markers are negative in BRBNS. In the latter, irregular dilated capillary spaces surrounded by a thin layer of endothelial cells (which are CD34-positive) can be identified in the dermis or subcutaneous adipose tissue.7The diagnosis of BRBN is confirmed by histopathological and immunohistochemical examination and a wide range of therapies have been described, from watchful waiting to surgical excision, laser therapy, sclerotherapy of lesions and systemic sirolimus. Treatment will vary according to the symptoms, organ involvement, and development of complications over the course of the disease, taking into account that patients with mild gastrointestinal bleeding can be managed conservatively with iron supplements and transfusions. On the other hand, patients showing greater severity are advised to start systemic treatment with sirolimus. Sirolimus acts by inhibiting angiogenesis and has shown good results.8The clinical similarities between these two conditions in the absence of histological study can lead to an incorrect diagnosis and, therefore, to inadequate treatment.None declared.Elsa Stella Mosquera-Belalcazar: Drafting and editing of the manuscript.Aline Alves Domingues: Review of the literature and drafting of the manuscript.Alessandra Coppinni: Review of the literature and drafting of the manuscript.Laura Luzzatto: Effective participation in data analysis; approval of the final version of the manuscript.Ana Elisa Kiszewski: Design and planning of the study; critical review of the manuscript and approval of the final version of the manuscript.None declared.


Schwannomas are rare peripheral nerve tumors that can present with diverse clinical manifestations. They commonly present as solitary, encapsulated masses and can occur in various locations throughout the body. This case report presents a comprehensive analysis of a peripheral schwannoma in a 29-year-old male patient who presented with numbness, pain, and paresthesias on the plantar aspect of the left foot. The symptoms progressively worsened, impacting the patient's daily activities. Physical examination revealed tenderness on the medial aspect of the left foot, along with prolonged episodes of paresthesia and recurrent numbness. Imaging studies confirmed the presence of a retro-malleolar mass, consistent with a peripheral schwannoma. The patient underwent successful surgical excision of the mass, resulting in complete resolution of symptoms. This case emphasizes the importance of considering peripheral schwannomas in the differential diagnosis of patients presenting with foot symptoms and highlights the effectiveness of surgical excision as a treatment modality for these tumors. Schwannomas, also known as neurilemmomas, are benign tumors originating from the Schwann cells that surround the sheaths of peripheral, cranial, or autonomic nerves. These encapsulated tumors develop when proliferating Schwann cells form a tumor around the nerve sheath . SchwannStandard X-rays typically yield negative results for soft tissue tumors. Ultrasonography and magnetic resonance imaging (MRI) play crucial roles in diagnosing schwannomas, particularly in evaluating superficially located tumors. Schwannomas generally display an iso-intense signal or reduced signal intensity compared to skeletal muscle on T1-weighted images, whereas they demonstrate heterogeneously increased signal intensity on T2-weighted images. Moreover, magnetic resonance imaging helps determine the size, shape, edge, surrounding edema of the lesion, invasion of adjacent blood nerves, and changes in signal intensity, aiding in accurate diagnosis ,4.The decision to perform surgery for benign neurogenic tumors should be based on a careful assessment of the risks and benefits. Surgical removal is essential in cases of large tumors that affect major peripheral nerves in the extremities and when there are accompanying symptoms of compression. In the foot, even small schwannomas affecting the plantar aspect can cause symptoms during walking and jumping. Surgical treatment usually includes the removal of the lesion through excision or intracapsular enucleation after making an incision in the epineurium. This surgical approach enables the preservation of the parent nerve and its neurological function, facilitated by the schwannoma's eccentric location . This caA 29-year-old male patient with no significant past medical history presented to the orthopedic department of a tertiary care hospital with a six-month history of numbness, pain, and paresthesias affecting the plantar aspect of his left foot. The symptoms gradually worsened over time, significantly impacting his daily activities. The patient reported experiencing pain specifically while walking on the medial aspect of his left foot. Additionally, the paresthesia and numbness episodes were lasting longer and occurring with increasing frequency.On examination of the left foot, a localized swelling was observed behind the medial malleolus, which was firm and non-tender to palpation with a positive Tinel test. The skin appeared normal, without any discoloration or visible lesions. The patient reported numbness and paresthesia in the distribution of the left tibial nerve, extending to the sole, heel, and toes. Sensory testing with a monofilament confirmed decreased sensation in the affected areas compared to the unaffected foot. Motor examination was suggestive of normal findings, completely. The patient demonstrated good balance and coordination during gait and balance assessment. Vascular examination revealed palpable dorsalis pedis and posterior tibial pulses, indicating normal arterial blood supply to the foot.The initial differential diagnoses considered for the patient's clinical manifestations included peripheral neuropathy, tarsal tunnel syndrome, Morton's neuroma, lipoma, ganglion cyst, vascular malformation, and posterior tibial schwannoma.To further investigate the patient's condition, various diagnostic modalities were employed. Ultrasound imaging revealed an oval-shaped hypoechoic solid mass measuring 3.1 × 1.6 cm near the medial malleolus, raising suspicion of a benign peripheral nerve sheath tumor . Fine needle aspiration cytology (FNAC) was performed under ultrasound guidance to obtain tissue samples from the mass. Four unstained slides, including two Giemsa and two Pap-stained slides, were prepared for microscopic examination. Microscopic examination of the smears revealed scattered fragments of a mesenchymal neoplasm consisting of fusiform spindle cells. The cells exhibited elongated, darkly stained nuclei and pale cytoplasm. Notably, no mitotic activity, cytologic atypia, or necrosis was observed. These findings were consistent with a spindle cell neoplasm.Magnetic resonance imaging (MRI) with contrast revealed a 19 × 20 × 28 mm intensely enhancing solid ovoid lesion over the posterior aspect of the distal tibia along the posterior tibial neurovascular bundle, approximately 39 mm from the superior surface of the calcaneus  were specifically identified as schwannomas situated in the lower extremity. Another study by Odom et al. estimated the prevalence of schwannomas in the foot to be as low as 2.93% [The onset of symptoms is associated with progressive tumor growth and the development of a sufficiently large tumor volume that compresses the originating nerve. Prolonged compression initially leads to venous insufficiency, causing fatigue, discomfort, and eventually pain in the affected area. Mechanical compression also disrupts axonal transport, exacerbating nerve dysfunction and resulting in structural damage, axonolysis, and Wallerian degeneration -6. CliniIn terms of diagnostic modalities, ultrasonography typically reveals solid, sharply delineated, ovoid, hypoechoic homogenous masses. X-rays may be performed to rule out any bone involvement or abnormalities. Further evaluation can be conducted using magnetic resonance imaging (MRI) with gadolinium contrast. Schwannomas generally display an iso-intense signal or reduced signal intensity compared to skeletal muscle on T1-weighted images, whereas they demonstrate heterogeneously increased signal intensity on T2-weighted images. Certain imaging features such as the "fascicular sign," "target sign," and "split fat sign" may be observed, although some of these signs can also be seen in neurofibromas. Large schwannomas with heterogeneous appearances on MRI can be distinguished from malignant peripheral nerve sheath tumors by analyzing the imaging features at and near the margin of the mass. The presence of the split fat sign and bright rim sign, along with the absence of a lobular shape and extensive edema, favors the diagnosis of schwannoma, particularly when two or more of these imaging features are present ,8. UltraHistopathologically, schwannomas can be classified into two distinct types known as Antoni A and Antoni B. Antoni A primarily consists of spindle cells with ill-defined boundaries, arranged in leaves or bundles, and exhibiting wavy nuclei. Mitotic figures are sparsely distributed. Verocay bodies, characterized by palisade arrangements of cells with areas devoid of nuclei between them, are also observed. On the other hand, Antoni B regions are less cellular and lack distinct architectural features. The matrix in Antoni B areas contains a mixture of spindle and oval cells, numerous blood vessels, sparse collagen, and a variety of inflammatory cells, including histiocytes. Some histiocytes within these regions may contain hemosiderin. Degenerative changes, such as hemorrhage, vessel wall hyalinization, cyst formation, fibrin deposition, focal calcification, and matrix fibrosis, are commonly observed. Schwann cell nuclei become hyperchromatic and multilobulated, resulting in a grayish-yellow coloration. Immunohistochemical tests show positive immunoreactivity for the S-100 and Leu-7 proteins [Treatment modalities for major nerve schwannomas depend on the patient's symptoms. Asymptomatic patients with confirmed diagnoses through MRI are typically managed conservatively. However, it has been cautioned that larger tumors carry a higher risk of major neurological deficits following surgery, suggesting that early surgical enucleation is strongly recommended when a schwannoma is detected. Surgical enucleation is the established treatment modality, although some schwannomas may pose challenges for complete enucleation, potentially leading to iatrogenic nerve injuries despite atraumatic procedures. Adequate exposure of the affected nerve, with sufficient proximal and distal margins, is crucial for precise tumor visualization, intraoperative nerve stimulation, and monitoring. The use of intraoperative magnifying glasses and gentle nerve manipulation helps preserve nerve continuity and function. Patients should be informed about the possibility of transient partial sensory or motor function loss in the affected nerve region .The diagnosis of schwannoma in the lower limb often experiences a delay of several years due to frequent misdiagnosis as a benign solitary mass, such as a myxoma, fibroma, or ganglion . The priSchwannomas typically exhibit slow growth and manifest as painless swelling that persists for several years without specific symptoms. The diagnosis of a schwannoma in a lower limb is often delayed due to frequent misdiagnosis as a benign solitary mass, such as a ganglion, fibroma, or myxoma. Recurrence is extremely rare, occurring in less than 1% of cases, unless there is incomplete excision of the tumor . MalignaSchwannoma of the foot is a rare neoplasm that affects both males and females equally. Histologically, it is characterized by the presence of palisaded spindle cells forming distinct Antoni A and B zones. Ultrasonography and magnetic resonance imaging play crucial roles in diagnosing schwannomas, providing valuable information regarding tumor location, size, composition, involvement of adjacent nerves, and relationships with surrounding muscular structures. Surgical excision is the primary treatment modality, and recurrence is infrequent .This case report highlights the clinical presentation, diagnostic workup, and successful treatment of a peripheral schwannoma involving the plantar aspect of the left foot. Peripheral schwannomas should be considered in the differential diagnosis of patients presenting with foot symptoms, and appropriate imaging studies, such as ultrasound and MRI, can aid in the diagnosis. Surgical excision remains the primary treatment modality, leading to favorable outcomes and symptom resolution. Healthcare professionals should be aware of peripheral schwannomas as a potential cause of foot symptoms, ensuring timely diagnosis and appropriate management.


Rosai–Dorfman disease is a benign lymphatic tissue proliferative disease with slow growth and is rarely observed in the clinic. Most of these patients present clinically with enlarged lymph nodes. In patients with spinal extranodal Rosai–Dorfman disease, which is even rare than the disease of lymph nodes, patients may experience numbness and weakness in the extremities.We report a 32-year-old male patient with multi-segmental spinal Rosai–Dorfman disease. On admission, his left fingers had been numb for 2 months. Over a 2-month period, the limb numbness progressed from the left to the right hand. The patient underwent resection of the lesion and internal fixation of the C2–C7 spine. The postoperative outcomes were satisfied and no recurrence was observed at 1-year follow-up.Spinal Rosai–Dorfman disease is a relatively rare tumor of which the pathogenesis is still unclear, and most patients have no specific clinical manifestations. Characteristic imaging findings can indicate this disease, however, a definitive diagnosis still depends on a pathological examination. Currently, total surgical resection of the tumor is a relatively effective and preferred treatment. This kind of tumor can occur at different sites, but usually in lymph nodes. RDD mostly presents as subcutaneous, isolated facial lymph node enlargement and is usually confined to the lymph nodes. RDD originating outside of the lymph nodes is rare, and a primary origin in the spine is even rarer. Most of these patients present clinically with symptoms of compression of the corresponding segment of the spinal cord, such as pain and numbness in the extremities or trunk. The severity of most clinical manifestations is related to the location of the primary tumor and the size of the tumor.A 32-year-old man presented with numbness of the extremities for 2 months before admission to hospital. The patient began to experience numbness in his left fingers in March 2021 without any apparent cause, which gradually progressed to the palm, the right upper limb, and finally, the trunk. He also had unsteadiness in holding objects in both hands, difficulty in performing fine movements, and pain in the back of his neck. These symptoms were worse at rest and could be relieved by activity. The patient had no nausea, vomiting, chest sulking, shortness of breath, cough, sputum, or other discomforts. He denied a history of hereditary disease in his family. He was admitted to the Department of Neurosurgery of the First Affiliated Hospital of Shenzhen University because of worsening symptoms. A physical examination showed decreased shallow and deep sensations in the left upper limb and decreased shallow sensation in the left thoracoabdominal wall (T2–L2 level). Routine blood laboratory tests, such as a full blood count, and renal and liver function tests, were within the normal range.Magnetic resonance imaging (MRI) of the spine showed that the mass was 45 × 23 × 46 mm in size. There was hypointensity on T1-weighted imaging (T1WI) and hypo- to isointensity on T2WI of an intradural extramedullary tumor at the C2–C5 level. After intravenous administration of gadolinium, the mass showed obvious enhancement and a tendency to compress the spinal cord anteriorly, without bone and soft tissue destruction. The bone signal of the C2–C5 spinous process was abnormal, the number of cervical lymph nodes was increased, and some lymph nodes were slightly enlarged. The diagnosis of potential eosinophilic granuloma at the C2–C5 level was made before surgery on the basis of the above-mentioned imaging findings . The posterior median line of the C2–C7 spinous process was precisely positioned as the surgical incision by combining preoperative MRI, body surface markers, and a C-arm. A line for surgical incision was routinely drawn, disinfected, and toweled, and the posterior median approach was used. The skin and subcutaneous and deep fascia were dissected, and the muscles were separated on both sides along the spinous process and the vertebral plate on the lateral side. A microscopic examination revealed a yellowish, tough, blood-rich, irregular mass with poorly defined borders in the muscle adjacent to the C2–C6 spinous process. There was also partial destruction of the C2–C6 spinous process and vertebral plate bony structures. After microscopic total excision of the paravertebral tissue mass outside the spinal canal, the mass was sent for intraoperative frozen biopsy. The biopsy showed that the cells of the tumor were short and spindle-shaped, and there was no obvious heterogeneity. Additionally, some plasma cells and lymphocytes and neutrophil infiltration were observed around the tumor. The C2 and C7 muscle attachment points were stripped during the operation. In order to maintain patient postoperative cervical stability, the C2 pedicle screw and C3-C7 lateral mass screws were implantated. The epidural mass was totally removed from the spinal canal, and the dura mater was left intact. The tumor was carefully removed from the edge of the dura mater. After removal of the epidural mass, a distinct pressure mark was visible on the dura. When the C2–C7 dura was sliced using microscopy scissors, a yellowish subdural mass was immediately observed in the dorsal position under the extramedullary dura. The mass was carefully separated from the dura because it was not firmly attached and was totally removed. The dura mater was sutured tightly, and the C2–C7 segment was stabilized with plastic peptide rods before placing the first drainage tube, and the incision was closed. Throughout intraoperative neurophysiological monitoring, there were no abnormalities in movement or sensation , extranodal, and mixed types depending on the site of occurrence. In summary, the pathogenesis of RDD is unclear and needs to be confirmed by further research.Although the etiology of RDD is unclear, recent studies have shown that its occurrence is associated with a clonal origin. Therefore, RDD should be considered as a tumor rather than an inflammatory proliferative response. In our case, the patient had bilateral lymphadenopathy. When lesions occur in the spine, patients with RDD mainly show clinical manifestations of pinprick sensation, pain sensation, or hypesthesia and even paraparesis. The clinical manifestations of these patients are mostly related to the tumor growth site. Our patient presented with hypesthesia of the left limb.RDD is mostly cervical, massive, painless, peripheral, bilateral lymphadenopathy, and most tumors are painless with progressive enlargement.The imaging features of RDD are hypointensity on T1WI and T2WI and hyperintensity on T1WI with enhancement. Fifty percent of RDD cases present with multiple subcutaneous nodules that can be misdiagnosed as macula or papules.,16MRI is the most commonly used imaging examination for RDD. However, there are also no specific MRI features indicative of RDD. MRI scans show that the tumor signals generally present as a homogeneous, isointense mass with clear borders and have obvious enhancement after contrast administration according to T1WI. However, on T2WI, these lesions appear to be heterogeneously hypo- to isointense. These findings are similar to those in our case. Pathological results are the gold standard for diagnosis, and the immunohistochemical characteristics of RDD include S100 (+), CD68 (+), and CD1α (−). There is limited evidence of the effectiveness of chemotherapy because of the rare incidence of RDD. Some studies have reported that tumor necrosis factor-α  and other immunotherapies may be useful for systematic RDD.Because RDD is considered a benign disease, in cases without involvement of the central nervous system, conservative treatment is effective in 90% of patients. With regard to spinal RDD, surgical resection is still considered, because in addition to achieving complete resection and central nervous system decompression, mass for histological diagnosis can be obtained during surgery.RDD is a benign proliferative disease, and early surgical excision provides the best prognosis for patients. The findings in our case show that surgical treatment alone is effective for treating cervical spine extranodal RDD without the need for postoperative radiotherapy and chemotherapy. Even if the tumor involves the spinal canal, it can still be removed surgically. Based on our experience, when tumors involving subdural region, in order to reach total resection, we suggest cutting the dura even if there is a high risk of postoperative leakage of cerebral spinal fluid, therefore, close sutures should be preferred after surgery to effectively avoied leakage of cerebral spinal fluid. In lesions involving multiple spinal segments, intraoperative internal fixation can be performed, which can reduce postoperative complications, such as spinal instability.We thank the patient and his family for participating in this case report.Conceptualization: Zhengwen Kang.Data curation: Jianwei Chen, Chang Cai.Project administration: Zhengwen Kang.Writing – original draft: Xianfeng Rao, Jianwei Chen.Writing – review & editing: Bo Wang, Qiusheng Zhang.


Osseous metaplasia is an extremely rare occurrence in traditional serrated adenoma (TSA). We report a case of a 50-year-old female with a TSA with osseous metaplasia (OM). The adenoma was identified during a colonoscopy for endoscopic mucosal resection of a previously identified polyp. The polyp location was the rectum. The colonoscopy was negative for any signs of concurrent malignancy. This case report is the fifth case of OM in a TSA reported in English. The clinical significance of OM is uncertain, and there is limited literature describing these lesions. Traditional serrated adenomas (TSA) account for less than 1% of colorectal polyps and 1%-7% of all serrated lesions . OM was first described in gastric adenocarcinoma by Gruber in 1913, followed by the description of OM in four cases of colonic adenocarcinomas by Dukes in 1939 . While OA 50-year-old Caucasian woman was referred for colonoscopy for endoscopic mucosal resection of a previously identified polyp. Her past medical history includes gastroesophageal reflux disorder (GERD), hypertension, and hypothyroidism. One polyp with a diameter of 3.5 cm was resected from the rectum.A biopsy of the tissue revealed a large high rectal adenoma, particularly a TSA with OM. The polyp demonstrated a villiform growth pattern with pseudostratified eosinophilic columnar cells and dark, penicillate dysplastic nuclei. Slit-like seriations and foci of ectopic crypts were noted. There was no observed loss of polarity, nuclear enlargement, pleomorphism, glandular crowding, or malignant glands with desmoplastic reaction excluding high-grade dysplasia or invasive carcinoma. Colonoscopy revealed an otherwise healthy colon without additional polyps. No other metaplastic changes were noted by staining with hematoxylin and eosin (H&E) including squamous or clear cell metaplasia. The images of the histology slides can be seen in Figure OM is a type of mesenchymal metaplasia that constitutes a rare phenomenon for gastrointestinal tract (GIT) neoplasms. It is defined by the presence of benign bone tissue that forms in a location where it would not normally be found. It is described more commonly for colorectal carcinomas and their metastases. OM in benign lesions, particularly in TSAs, is an extremely rare finding with less than five reported cases Table . The clinical significance of OM in GIT neoplasms remains uncertain. Previously, OM was described to be associated with symptomatic bleeding and was considered a high-risk feature . In our For the previously reported four cases, the sites of TSA were the left colon (3/4) and rectum (1/4). In this case, the location of TSA was the rectum. The malignant transformation with the development of adenocarcinoma occurred in one of the four reported cases . For ourThe exact mechanism underlying OM is not fully understood, but several hypotheses exist in the literature. The ability of fibroblasts to undergo a transformation into osteoblasts may constitute the basis of such metaplasia . The subThis case report describes a rare finding of osseous metaplasia (OM) in traditional serrated adenoma. To the best of the authors' knowledge and based on an extensive literature review, this is the fifth case of OM in this specific type of colorectal polyp. Although it is a benign finding, additional data analysis is needed to understand the exact pathophysiologic mechanism and clinical significance.


An isolated hydatid cyst of the spleen is a rare presentation of echinococcal diseases, especially in non-endemic areas where it may end up with unnecessary work-up and misdiagnosis. Here, we present the case of a 28-year-old female presenting with generalized abdominal pain, constipation, and early satiety who had a delayed diagnosis of isolated splenic hydatid cyst which was partially treated with albendazole, eventually requiring splenectomy. Echinococcus, which belongs to the family Taeniidae. Cystic echinococcosis  is caused by E. granulosus, one of the most common species of Echinococcus [Echinococcal disease is caused by infection with the metacestode stage of the tapeworm nococcus . AlthougA 28-year-old Caucasian female with no significant past medical history presented to our facility with a two-week history of constipation, generalized abdominal discomfort, and early satiety. She reported backpacking for seven months  and had been to Europe, the Balkans, and Southeast Asia. She stated that she was staying at hostels and couch surfing frequently where she had animal contact including dogs and cats. She also reported eating street food frequently. She was seen by a provider one month prior with similar symptoms and was treated with empiric antibiotics for possible pyelonephritis. She reported that her symptoms failed to improve and had since progressively worsened which prompted her to present to our institution. Upon presentation to our institution, the physical examination was unremarkable except for tachycardia.On workup, complete blood count, comprehensive metabolic panel, and liver function tests were unremarkable. She underwent computerized tomography (CT) of the abdomen/pelvis with intravenous (IV) contrast, which showed a large cyst measuring 11.9 × 10.6 cm within the spleen pushing on the stomach  of the brain and abdomen/pelvis with and without contrast were negative for any other cystic lesions including any liver lesions Figure .Echinococcus given the patient’s travel history and the risk of anaphylaxis if any open surgical procedure was performed in the event of a hydatid cyst. After consultation with gastroenterology and infectious disease, we sent for echinococcal serologies and initiated the patient on albendazole 400 mg twice daily. While pending echinococcal serologies, the puncture-aspiration-injection-reaspiration (PAIR) procedure was done by interventional radiology (IR) with 500 cc of serosanguinous fluid aspirated from the splenic cyst. The pathology of the aspirate revealed scattered calcareous bodies, round structures with occasional granules, and a ring structure with hooklets highly suspicious of Echinococcus , the symptoms  would be expected to be more severe. Moreover, the rupture of a cyst can lead to an allergic reaction/anaphylaxis and even death. Not infrequently, the diagnosis is incidentally established when a patient is evaluated for other diseases . Our patEchinococcus.Confirmation of the diagnosis depends on imaging modalities, mostly abdominal ultrasonography and CT scan, serologies, and pathological evaluation. Calcification of the cyst wall and the presence of daughter cysts, cystic membranes, septa, or hydatid sand are imaging findings consistent with splenic hydatid disease. In most cases, serologic tests detecting anti-echinococcus antibodies and imaging characteristics in combination indicate the correct diagnosis ,8. DespiTreatment is mainly surgical and options depend on the individual patient and surgeon’s expertise. Total splenectomy, partial splenectomy, cyst enucleation, and unroofing with omentoplasty have all been reported . PercutaLimited data are available on epidemiology, clinical presentation, and establishing a diagnosis for an isolated splenic echinococcal cyst. Our case represents the importance of a high index of suspicion and clinical judgment in challenging diagnostic scenarios as recognition is critical given the risk of an anaphylactic reaction, potential death in the event of misdiagnosis, and open surgical or other unwarranted procedures. It also highlights the need for more research toward identifying the best diagnostic modalities in patients with isolated splenic echinococcal cysts.