Neobladder urolithiasis is a rare but important delaying complication of orthotopic urinary diversion. We report a case of Hem-o-Lok (HOLC) migration into the neobladder with giant stone formation after orthotopic neobladder cystectomy.We report a case of a 57-year-old man with frequent urination and occasional discharge of stones 3 years after a laparoscopic orthotopic neobladder cystectomy. Computed tomography revealed a large round 3.5 cm calculus. An endoscopic neocystolitholapaxy was performed, and a Hem-o-Lok was found in the center of the stone.We described the case presentation, treatment and analysis of etiology of stone formation to avoid such complication. Neobladder urolithiasis is a rare but important delayed complication of orthotopic urinary diversion after laparoscopic orthotopic neobladder cystectomy . Stone fA 57-year-old man with frequent urination and occasional discharge of stones from the urethra was admitted to The Affiliated Hospital of Qingdao University. A laparoscopic orthotopic neobladder cystectomy was performed 3 years ago, and the post-surgery histopathology showed T2a muscle invasive bladder cancer. He did not continue the oncological follow-up. The patient complained of frequent urination and occasional stone discharge from the urethra. A urinary routine test showed urinary occult blood 2+, urinary leukocyte 1+, bacteria count 435.60/µl, and nitrite -. Computed tomography (CT) of the abdomen and pelvis revealed (Fig. Laparoscopic orthotopic neobladder cystectomy is still the standard treatment for muscle invasive bladder cancer. In laparoscopic cystectomy, HOLC is used to ligate the vesical arteries, the vas deferens, the bilateral ureters, the seminal vesicle arteries, and the prostatic pedicles. Compared with titanium clips, HOLC have better histocompatibility and tolerance, and have less impact on CT and MRI examinations. However, due to its non-absorbability, HOLC occasionally cause tissue and organ perforation or migration. Shun-Fa et al. have reported a case of HOLC migrating into the bladder with stone formation after laparoscopic radical nephroureterectomy and bladder cuff excision . Li Wan The patient was treated with the intracorporeal Studer technique, a neobladder reconstruction with folded detubularized ileu . When deAnother concern is that HOLC migration into the neobladder can cause complications such as lower urinary tract symptoms, stone formation, or anastomotic fistula. Neobladder stones can be asymptomatic and discoverable incidentally on a radiological examination. However, with the enlargement of the stone, lower urinary tract symptoms are obvious. Tiu, A and Nguyen, S et al. respectively reported large neobladder stones of over 750 g , 7. The Hem-o-Lok clip migration into the neobladder with stone formation after laparoscopic orthotopic cystectomy is an extremely rare complication that has been rarely reported in the literature. In order to prevent the migration of the HOLC and its complications, the use of the HOLC should be reduced under the premise of ensuring the effectiveness, and the loose HOLC should be removed in time. Regular re-examination of urine routine to avoid urinary infection and intermittent self-catheterization to prevent the occurrence of urinary retention may be effective methods to prevent the formation of neobladder stones.


The use of intragastric balloons (IGBs) for weight loss has increased in popularity over the past three decades. While they are generally considered effective and safe, there have been reports of various complications ranging from mild to severe. Acute pancreatitis is a rare complication following IGB insertion. In this case report, we describe the occurrence of acute pancreatitis in a patient six months after IGB insertion . The balloon was found to be in the appropriate position and was removed endoscopically, leading to rapid clinical and biological improvement. The mos2 ,4. Here,2), for which she underwent IGB insertion  six months ago for weight loss (current BMI = 30 kg/m2). She denied any recent abdominal trauma or new drug use. Her family history was insignificant. The clinical evaluation revealed a mildly tachycardic patient, who was otherwise stable, and an isolated severe epigastric tenderness on abdominal examination. Lab tests showed mild leukocytosis without C-reactive protein elevation, normal renal and liver function tests, and an elevated lipase level four times the upper limit of normal (435 IU/L). The extended analysis showed serum calcium and triglyceride levels within the normal range. We performed an abdominal CT scan which showed an IGB that was over-distended with an approximate volume of 1,140 mL (14 × 11 × 14 cm), half-filled with fluid and displaying an air-fluid level. At the level of the pancreas, the body and tail were compressed by the IGB, with some fluid below the tail, suggesting mild acute pancreatitis  score = 0) secondary to extrinsic pancreatic compression by IGB. She was hospitalized and started on intravenous hydration, painkillers, and an antiemetic. Urgent upper gastrointestinal endoscopy was performed. The balloon was in the correct gastric position and was deflated and removed without complications. The gastric mucosa was inspected and intact. The patient began to improve immediately following the balloon removal. Her pain was relieved and she started to tolerate oral intake. She was discharged home on day two free of symptoms. The scheduled follow-up one week later was unremarkable.The technology of IGB has not stopped evolving since its invention four decades earlier. Currently, there are various types available in the market that differ in their characteristics  but sharAcute pancreatitis following IGB is rare. Only a few case reports were described in the literature before the Food and Drug Administration (FDA) issued a warning in 2016. Since the FDA warning was issued in 2016, the agency has received reports of over 30 cases of acute pancreatitis . The exaSimilarly, spontaneous hyperinflation has been also reported by the FDA as a potential complication of fluid-filled IGB. Patients should be informed and educated about this potential complication . Its exaAcute pancreatitis in the context of spontaneous hyperinflation of the fluid-filled balloons is a rarer presentation . PancreaOur patient developed a mild case of acute pancreatitis secondary to the IGB placement. This complication occurred at the end of the intended placement duration of six months, despite following manufacturer instructions. The balloon was in its correct gastric location but was found to be hyperinflated and containing an air-fluid level. The extrinsic compression of the pancreas by the distended stomach is believed to be the etiology of acute pancreatitis. The endoscopic removal of the balloon led to rapid clinical improvement and correction of the biochemical abnormalities. Despite their low incidence, clinicians should be aware of acute pancreatitis and hyperinflation as potential complications of IGB placement. Prompt management should be done to avoid severe consequences. The role of asymptomatic elevation in pancreatic enzymes as an early marker for the prediction of this complication should be explored in subsequent research.


An 81-year-old female patient underwent a screening mammogram one year after completing treatment for right-sided estrogen receptor (ER)/progesterone receptor (PR)-negative ductal carcinoma in situ (DCIS). A new 1-cm mass was noted in the contralateral breast. Ultrasound and percutaneous core needle biopsy results were suggestive of an atypical papillary lesion. An excisional biopsy was performed, and the final pathology was consistent with a benign adenomyoepithelioma (AME). Surgical resection was considered her definitive treatment. AME of the breast is a rare clinical entity, with only a handful of case reports and case series available. In this case report, we review common clinical and radiologic presentations, methods of diagnosis, and recommendations for management based on current literature. The presence of an AME in the background of a previous or synchronous breast malignancy occurs in a very small percentage of cases. On review of available literature, we identified other cases with a past or current history of breast malignancy. Adenomyoepithelioma (AME) of the breast is a rare tumor that is characterized by biphasic proliferation of both epithelial and myoepithelial cells . AlthougAMEs tend to present in patients over 50 years of age but have been known to occur in younger populations . InitialThe patient is an 81-year-old woman with a history of right-sided ER/PR-negative DCIS treated with lumpectomy and radiation one year prior who was found to have a new left-sided breast mass on screening mammography. Her diagnostic mammogram showed a 1-cm oval mass with indistinct margins in the central area of the left breast (Figure Ultrasound was performed for further evaluation and showed an ovoid circumscribed 1.2 x 0.8 x 0.8 cm hypoechoic mass with indistinct margins at the 9:00 position located 10 cm from the nipple in the left breast. The internal vascular flow was noted. Limited visualization of the left axilla showed morphologically benign lymph nodes (Figure Ultrasound-guided core needle biopsy was performed two weeks later. Pathology showed a focally monotonous papillary lesion consistent with an atypical papillary lesion. The specimen was negative for malignancy. The excisional biopsy of this mass was recommended for definitive diagnosis due to the atypia noted on core biopsy sampling. The patient underwent a localized excisional biopsy of the left breast. The specimen consisted of a gray-yellow firm granular mass with dimensions measuring 1.6 x 1.2 x 1 cm.Histopathological analysis of the surgical pathology showed adenomyoepithelioma without evidence of malignancy and negative margins greater than 1 cm from the mass. The patient was seen by the breast surgery clinic one month after the procedure for follow-up. She continues to do well with return to daily activities and reports no new health concerns. Given her good health status and recent abnormal breast findings, she elected to continue regular screening mammograms at this time and is scheduled for a bilateral diagnostic mammogram next year.AME of the breast is a rare neoplasm that was first described in 1970 by Hamperl et al.  and lateVariable clinical and radiological presentations represent significant challenges in diagnosis. Current literature suggests that some patterns are more common. AMEs are most likely to occur in the fifth to sixth decades of life . These tRadiologic presentations also tend to be widely varied. Because mammography and ultrasound are commonly used for first-line identification of breast masses, findings from these imaging modalities are the most well-studied. On mammography, findings are non-specific and can include masses, microcalcifications, and focal asymmetry . LesionsBecause clinical and radiographic findings are non-specific, differentiation of AMEs from other breast masses is difficult. Current literature notes that the most commonly observed features of AMEs include oval masses or irregular hypoechoic masses with circumscribed or microlobulated margins . Oval maAlthough the vast majority of patients with AME have no associated malignancy, both ipsilateral and contralateral AMEs have been described in patients with past or concurrent malignancies ,10. InteThe treatment of AME is debated among clinicians, and guidelines have yet to be established owing to the lack of prospective studies. Based on the current literature, complete surgical resection tends to be sufficient for benign AME, while malignant AME could require additional treatment with radiation or chemotherapy . CliniciAdenomyoepithelioma of the breast is a rare tumor that is most commonly benign but can undergo malignant transformation. Diagnosis of AME is challenging given its variability in clinical and radiologic presentation. Therefore, tissue pathology is required to make a diagnosis. Guidelines for management have not yet been established as the rarity of AME makes prospective studies difficult. Most clinicians recommend wide surgical excision for definitive diagnosis and treatment.


Retropharyngeal calcific tendinitis (RCT) is a relatively benign condition of calcination of the longus colli muscle tendon of unknown origin, which causes severe acute neck pain. However, it is often not recognised, which leads to delayed diagnosis and unnecessary treatment.We have searched PubMed and Google Scholar for publications which reported at least one patient with RCT and were published in the last 20 years. The literature was then analysed according to the PRISMA-S protocol. We also report three patients with RCT presenting at the Neurological Emergency Unit, University Medical Centre, Ljubljana, Slovenia, from 1 January 2020 to 1 June 2022. We discuss their clinical presentation and differential diagnosis, explain our decision-making process, and briefly describe the clinical course. Case reports have been performed according to the CARE protocol.We have analysed a total of 112 titles with 231 patients. The most frequent symptoms and signs were: neck pain, neck stiffness and odynophagia, as was the case in our reported cases.RCT is a dramatic yet self-limiting condition, often not recognised in time. An effort should be made to increase neurologists’ awareness about this condition. Retropharyngeal calcific tendinitis (RCT) is, according to ICHD-3 criteria, a “headache caused by inflammation or calcification in the retropharyngeal soft tissues”.1 It occurs as a result of poorly understood mechanisms in the upper fibres of the longus colli muscle. Data from a similar condition affecting the shoulder joint showed neovascularisation and new nerve growth as a result of the innate immune response.2 This often dramatic but self-limiting condition is often not recognised among physicians, which leads to unnecessary diagnostic procedures and delayed treatment.4 Besides neurologists, other specialists are also involved in treatment of this condition, especially otorhinolaryngologists (ENT)4 and orthopaedic surgeons.5 The annual incidence of RCT is estimated to be from 0.5 cases per 100 000, up to 1.1 case per 1000.6Calcifications in the retropharyngeal space as a cause of severe acute head and neck pain have long been recognised, but have only recently been incorporated in the latest . The analysis of the literature was performed according to the PRISMA-S protocol.8In addition, we report three cases of retropharyngeal calcific tendinitis (RCT) that were referred to the Neurological Emergency Unit (NEU), Division of Neurology, University Medical Centre Ljubljana, Slovenia, from 1 January 2020 to 1 June 2022. The reports follow the CARE protocol.NEU is a tertiary medical facility for over 300 000 inhabitants where 25 016 patients were examined in the time period described above, of which 2073 were discharged with the main diagnosis “Headache, unspecified” (G 44.8 or R 51) according to ICD-10 and only three were diagnosed with RCT (M 65.2).9/l). Although the patient was afebrile, the retropharyngeal abscess was still considered in the differential diagnosis. Magnetic resonance imaging (MRI) of the neck showed fluid collection and swelling in the cranial part of the longus capitis/colli muscle on the left  of 20 mg/l  and white blood cell count (WBC) of 10.2  of the aortocervical and intracranial vessels revealed no vascular abnormalities. MRI of the cervical spine revealed prevertebral oedema from the C1 to the C4 level , not seeIn May 2022, a previously healthy 43-year-old female experienced pain in the right posterior neck which evolved gradually over a period of 48 hours. She described an electric shock-like pain that was radiating to the occipital area at any attempt of head extension, and even if she tried to hold the head in the neutral position. Laboratory workup at the Medical Emergency Unit revealed only slightly elevated CRP. Pain medication (metamizole 2.5 g IV) was minimally effective. Three days after the onset of pain the patient was examined at the NEU where occipital neuralgia was suspected, and outpatient MRI of the head and neck was suggested. As the pain continued, she came to the NEU on day 5 and spondylodiscitis was added to differential diagnosis. A neck CT revealed calcifications in the region of alar ligaments and repeated laboratory results showed elevated CRP of 20 mg/l , with no other abnormalities. The patient was discharged with ibuprofen 1200 mg daily. As the pain continued without relief, she returned to the NEU on day 7. This time RCT was diagnosed and a short course of corticosteroids was prescribed . The pain disappeared within one week.The literature search returned no randomised controlled trials, meta analyses, clinical trials or systematic reviews. We retrieved a total of 198 titles (Online Resources- 1 and 2), all of which were case and series reports and reviews. Of those, 112 were eligible for the study. The details of article selection are given in 9 This at least partially explains occipital (headache) and pharyngeal (odynophagia) irradiation of the pain as well as neck stiffness (or decreased range of motion), as seen in our patients. Torticollis is sometimes reported, but may represent exaggerated neck stiffness. A detailed review of the (rare) causes of craniofacial and neck pain can be found in the literature.10According to ICHD-3 criteria, RCT represents a “headache caused by inflammation or calcification in the retropharyngeal soft tissues”. The pain is usually severe, continuous, throbbing or electrising in quality. Trigeminal afferent fibres from dura and cervical afferent fibres from the skin and muscular tissue in the cervical region converge to synapse onto the same second-order neurons in the trigeminocervical complex.11 Immunological mechanisms involving the innate immune system in the form of new nerve growth and neovascularisation within the tendon in calcific tendinitis of the shoulder joint have been reported.12 Similar processes in the retropharyngeal space may be a plausible explanation in patients with RCT. However, further studies are needed to confirm these speculations.Little is known about the causes of calcium deposition or inflammation in the longus colli muscle. One report on histological findings in the retropharyngeal tissue of RCT patients has revealed a foreign-body type of inflammation around hydroxyapatite crystals.Differential diagnosis includes other similar conditions that should be promptly recognised by neurologists .1, not typically seen in RCT. They are also accompanied by neck stiffness, but only on flexion, not on extension or rotational movements.1Meningitis and meningoencephalitis are usually accompanied by fever, photophobia, nausea and/or vomiting, or even an altered mental state9/l.13 Spondylodiscitis, another possible cause of acute severe neck pain, is usually accompanied by clinical and laboratory signs of systemic inflammation, and diagnosis is made after appropriate imaging.15 Spontaneous carotid or vertebral artery dissection could present as an isolated head and/or neck pain, sometimes accompanied by nausea, but no neck stiffness has been reported.16 Lastly, greater occipital nerve (GON) neuralgia and cervicogenic headache (CH), which are relatively benign conditions, characterised by prominent neck pain, should also be kept in mind1.17 However, pain in GON neuralgia emerges in the form of short (seconds to minutes) attacks, leaving tenderness or allodynia in the region of GON.1 Therefore, when pain evolves over a period of several hours or even days, other causes should be considered. CH is a broad category of chronic conditions, with some diagnostic criteria overlapping with RCT.Odynophagia is often seen in RCT, but is a hallmark of retropharyngeal abscess, where in most cases, the blood count shows elevated leucocytes above 12 × 10Radiologically, differential possibilities in patients with suspected RCT include retropharyngeal abscess, tumour or even trauma; therefore, familiarity with typical imaging findings facilitates early diagnosis and may prevent inappropriate therapeutic procedures .18 CT reliably confirms the location of calcifications within the superior fibres of the longus colli tendons and may show soft tissue swelling and/or small retropharyngeal effusion as well.19 Nowadays, the first imaging modality that these patients undergo is usually MRI, which demonstrates oedema and retropharyngeal effusion clearly. However, as calcifications are much subtler on MRI in comparison to CT, a high level of clinical suspicion is needed for the correct diagnosis. On the other hand, the presence of peripheral postcontrast enhancement or the evidence of pus on diffusion-weighted imaging should suggest infection as the cause.14 CT is immediately available and cheap but exposes the patient to ionizing radiation, which may be inappropriate in women in the childbearing age. MRI is not as accessible as CT, is expensive, and lasts longer, which may pose a problem due to inability of the patient to lay still in the supine position. Finally, MRI may be contraindicated in the cases of metallic implants or claustrophobia.As plain film radiography may miss subtle calcifications within the tendon, CT is the preferred imaging modality due to its supreme resolution and multiplanar capabilities.RCT is a rare disorder that neurologists should be familiar with. It is also a relatively new subject for neurologists: it has been recognised as a neurologic disorder only in 2013, when the ICHD-3 criteria were published. Typically, the patient is a previously healthy middle-aged person, presenting with the triad of neck pain, neck immobility and painful swallowing. Mildly or moderately elevated CRP and leucocytes are usually present. Neck MRI and/or CT imaging should be performed to confirm diagnosis. Treatment with oral steroids or non-steroidal anti-inflammatory drugs should lead to prompt symptom alleviation.


Pelvic lipomatosis is a proliferative disease characterised by excessive fat growth in retroperitoneal space leading to inadequate bladder drainage and ureteral compression. Cystitis glandularis, cystitis cystica, or cystitis follicularis can be found in the majority of patients with the disease.We report a case of a 63-year-old man diagnosed outside our hospital with pelvic lipomatosis after finding a pelvic mass behind the bladder causing severe bilateral hydronephrosis. A bladder-sparing excision of the pelvic lipomatosis mass with bilateral ureteric reimplantation was performed, thereby avoiding the need for urinary diversion. Our case supports the hypothesis that pelvic fat mass extirpation and ureteral reimplantation is an effective surgical treatment strategy for pelvic lipomatosis. In the latter half of the 1950s, Engles discovered a rare condition called pelvic lipomatosis now. The disease is proliferative, characterised by excessive fat growth in the retroperitoneal space of the pelvis .There is limited research on the disease itself and its long-term effects, and so there are no set guidelines for patients to follow. There are numerous treatment choices, such as watchful waiting or surgery that may involve a total cystectomy or urinary diversion. There have been recent reports of success found in a bladder-sparing technique .We report a case of a 63-year-old man diagnosed outside our hospital with pelvic lipomatosis after finding a pelvic mass behind the bladder causing severe bilateral hydronephrosis. A bladder-sparing excision of the pelvic lipomatosis mass with bilateral ureteric reimplantation was performed, thereby avoiding the need for urinary diversion. Our case supports the hypothesis that pelvic fat mass extirpation and ureteral reimplantation is an effective surgical treatment for pelvic lipomatosis. This article was previously posted to the Research Square preprint server on June 13, 2023.A 63-year-old African male presented with a chronic history of lower abdominal pain for more than four years not associated with lower urinary tract symptoms. The patient was diagnosed outside our hospital with pelvic lipomatosis after finding a pelvic mass behind the bladder causing severe bilateral hydronephrosis. He underwent partial pelvic mass excision with bilateral double J stent insertion last year. The patient was referred to our urology department for a second opinion. Histopathology slides were re-submitted and confirmed the diagnosis. His laboratory investigations showed creatinine 109 µmol/L.The abdominal and pelvic CT scan showed a pear-shaped elongated urinary bladder secondary to extensive pelvic lipomatosis Figure . The finThe patient underwent cystoscopy which showed an elongation of the prostatic urethra with a high bladder neck. There were several bullous lesions over the bladder neck and trigone without the involvement of the ureteric orifices. A tissue biopsy of the lesions demonstrated cystitis glandularis Figure .A laparotomy was performed through a midline incision, and the bladder was found high reaching intraperitoneal space Figure . BilaterPostoperatively, the patient was ambulated and started on an oral diet on postoperative day 2. Repeated CT showed marked improvement in bilateral hydronephrosis Figure .Unfortunately, on post-operative day 10 patient was arrested after he was diagnosed with a massive pulmonary embolism and passed away despite being on a prophylactic dose of anticoagulant medications.Pelvic lipomatosis is an uncommon disease associated with an overgrowth of fat in the pelvis and rectum areas . CystitiAdequate monitoring and follow-ups are necessary for patients with cystitis cystica or glandularis so that bladder adenocarcinoma can be detected if present and transurethral resection can be repeated . ClinicaThere is limited research on the disease itself and its long-term effects, and so there are no set guidelines for patients to follow. This also means that there are numerous choices of treatment, such as watchful waiting or surgery that may involve a total cystectomy or urinary diversion . TreatmeAccording to Carpenter AA, patients who have pelvic lipomatosis can be categorized according to age, as the disease is typically indolent in patients who are older . For oldFor patients suffering from pelvic lipomatosis and bilateral hydronephrosis, relief of obstructions to the urinary tract should be prioritized. This can be achieved through traditional surgery, such as a ureteral stent insertion, or more radical surgical options such as a total cystectomy and urinary diversion. There have been recent reports of success found in bladder-sparing techniques using mass extirpation and ureteral reimplantation.


An Oriental Shorthair cat, aged 1 year and 6 months, developed progressive stridor and a palpable right ventral cervical mass. Fine-needle aspiration of the mass was inconclusive, while thoracic radiography and CT showed no evidence of metastasis. There was initial improvement in stridor with oral doxycycline and prednisolone treatment, but it recurred 4 weeks later and excisional biopsy was performed. Histopathology with immunohistochemistry diagnosed leiomyosarcoma with incomplete surgical margins. Adjunctive radiation therapy was declined. Repeated physical examination and CT 7 months postoperatively documented no evidence of mass recurrence.This is the first reported case of retropharyngeal leiomyosarcoma in a young cat with no evidence of local reoccurrence 7 months following an excisional biopsy. They are frequently non-encapsulated and invasive tumours. Histological features vary from densely packed, relatively homogeneous spindle cells with the appearance of smooth muscle to more pleomorphic ovoid or round cells.Leiomyosarcomas are uncommon, malignant tumours arising from smooth muscle cells216Surgery is the recommended treatment of choice and the prognosis depends on the anatomical location of the tumour and the presence of metastasis.To the authors’ knowledge, this report describes a novel diagnosis of retropharyngeal leiomyosarcoma in a young cat with a good outcome following marginal surgical excision. This tumour should be considered as a differential diagnosis for a retropharyngeal mass in the cat.A neutered male Oriental Shorthair cat, aged 1 year and 6 months, was seen by the primary care practice (PCP) with stridor that had progressively worsened over the course of 3 weeks. Clinical examination was unremarkable. Haematology and serum biochemistry were within normal limits. The patient was sedated and examination of the oral cavity revealed a large retropharyngeal mass . A fine-The BAL results were consistent with mixed eosinophilic inflammation with suspected allergic disease. Fine-needle aspiration (FNA) results were inconclusive. The cat received a subcutaneous injection of dexamethasone .CT of the head and thorax were performed by the PCP and interpreted by a diagnostic imaging specialist. CT revealed a discrete right retropharyngeal/tonsillar mass with no local infiltration into the surrounding tissues, ipsilateral medial retropharyngeal lymphadenopathy and unspecific pulmonary changes .The cat was referred to Wear Referrals, Stockton-on-Tees, UK, and an approximately 1 × 2 cm firm soft tissue mass was palpated on the right lateral neck immediately ventral to the right tympanic bulla and cranioventral to the wing of the atlas, displacing the larynx to the left. The owner reported that the clinical signs were stable. The cat had moderate stridor but no other abnormalities on clinical examination. Owing to the cat’s young age, lack of conclusive evidence of neoplasia and the possibility of an inflammatory diagnosis, the cat was prescribed doxycycline  and prednisolone  for 4 weeks. The cat was re-examined 4 weeks later and the stridor had improved, although the mass was unchanged in size. Medications were continued for a further 5 weeks. On re-examination, the stridor had recurred and excisional biopsy was recommended.The cat was premedicated with medetomidine  and butorphanol . Cefuroxime  was administered 30 mins prior to surgery. General anaesthesia was induced using alfaxalone . Intubation was achieved using a 3.5 mm endotracheal tube, and anaesthesia was maintained with isoflurane  in 100% oxygen. Analgesia was provided with fentanyl  as a constant rate infusion.The patient was positioned in dorsal recumbency, with the neck extended and supported by a sandbag. A ventral midline cervical incision was created, and a combination of blunt and sharp dissection through subcutaneous tissue and sphincter coli muscle was performed to identify the mass ventral to the right tympanic bulla. The mass was marginally dissected from the local neurovascular structures , muscles  and other adjacent anatomy  using a combination of delicate, blunt and sharp dissection and bipolar electrosurgery. Lavage and routine closure were performed. The mass was submitted for histopathology.Intravenous fentanyl was discontinued immediately postoperatively, and the cat received buprenorphine . The cat was discharged 18 h postoperatively with prednisolone  for 4 weeks with a tapering dose and buprenorphine  for 3 days. The patient was reviewed 4 weeks following the surgery. The owner reported that the stridor had fully resolved.  of the head and thorax was performed. CT revealed no evidence of primary mass recurrence , static This report describes the first case of retropharyngeal leiomyosarcoma in a young cat and should be considered as a differential diagnosis for a mass in this location.The main presenting clinical sign was stridor, likely due to the partial obstruction of the upper respiratory tract and displacement of the larynx, and the mass could be readily identified by palpation and direct visualisation. followed by a 4-year-old cat with documented primary bladder leiomyosarcoma. While most of the reported cases feature middle-aged to older cats,15 the cat in this report was 1 year and 6 months old, which represents an uncommon, early presentation for this type of tumour.As far as we are aware, the youngest cats diagnosed with leiomyosarcoma were one cat aged 3 years and 9 months, which was diagnosed with oesophageal angioleiomyosarcoma,The initial lack of a definitive diagnosis by FNA, the young age of the cat and the findings of eosinophilic pulmonary disease influenced the initial decision to provide a therapeutic trial with oral antibiotics and steroids. The initial improvement in clinical signs may be due to reduced peritumoral inflammation. The failure of the mass to respond to treatment then prompted the decision for a more invasive procedure. Excisional biopsy was elected over Tru-cut or incisional biopsy as the clinical signs of stridor were likely due to the mass effect on the trachea and therefore a planned marginal excision would provide therapeutic benefit. The oesophagus of the cat appeared radiologically normal and would only be expected to contain smooth muscle in the intrathoracic portion. It is possible that this tumour may have arisen from vascular smooth muscle in this area. An additional differential diagnosis considered in this case was laryngeal tumour or cyst. Oral examination and CT ruled out a laryngeal origin of this mass.Immunohistochemistry was necessary because the histopathological features of the mass were similar to rhabdomyosarcoma and fibrosarcoma. The location of the mass, particularly in a young cat, also meant that other differential diagnoses would be more likely than leiomyosarcoma. Leiomyosarcomas that develop in the haired skin and subcutaneous tissues are thought to arise from smooth muscle associated with the vasculature or arrector pili muscles.Postoperatively, the cat was prescribed prednisolone to decrease postoperative inflammation and respiratory tract obstruction following the extensive dissection in the area, and because eosinophilic lung disease had previously been noted on BAL . It is unclear whether the treatment with prednisolone had any effect on the outcome, although corticosteroids are not generally considered to have antineoplastic action on mesenchymal tumours. Adjunctive radiation therapy has not been extensively reported for incompletely excised feline leiomyosarcomas, although it would be considered a beneficial therapy for residual microscopic disease. However, radiotherapy was declined by the owners. to 48 months. Numerous case reports documented the short-term prognosis and no tumour reoccurrence 5, 615 and 10 months postoperatively, respectively. At the time of writing, the cat had shown no signs of tumour recurrence; the presence of mild unilateral retropharyngeal lymphadenopathy and cranial mediastinal lymphadenopathy could indicate metastatic disease, although leiomyosarcoma do not typically metastasise via the lymphatic route. Other causes of the imaging findings  were considered more likely. Continued surveillance of the cat and, ultimately, post-mortem examination are required for complete information.Long-term follow-up data for cats treated for leiomyosarcoma are largely lacking. The reported prognosis following the surgical removal of the tumour varies from 1 monthLeiomyosarcoma should be a differential diagnosis for cats presenting with a retropharyngeal mass. A good medium-term outcome can be achieved with marginal excision.


Lipomas are benign tumors composed of adipose tissue that can occur in various locations throughout the body, including the gastrointestinal (GI) tract. Lipoma of the small bowel is a rare clinical condition. It infrequently results in small bowel obstruction and volvulus. In most of the patients, it is an incidental diagnosis. However, it may present with abdominal pain, nausea, vomiting, GI bleeding, and constipation. It is commonly diagnosed using imaging studies, such as computerized tomography scan or magnetic resonance imaging. The management of small bowel lipomas depends on the presence of symptoms and the risk of complications. Asymptomatic lipomas can be safely monitored with imaging studies, as the risk of complications is low. However, in symptomatic cases, surgical or endoscopic treatment may be necessary to relieve obstruction and prevent complications. We report a case of volvulus of small bowel in a middle-aged female presenting to our hospital with acute GI symptoms. The incidence of intestinal lipomas varies from 0.0035% to 4.44%. Small bowel lipomas are benign adipose growths, usually found within the bowel wall or mesentery of the small intestine, and are rarely encountered . IntestiA 47-year-old female was admitted to the emergency department with complaints of abdominal pain and inability to pass stool for five days and nausea associated with vomiting of around eight to ten episodes per day. The patient was on treatment for dyspepsia and constipation for the last one month. The patient had a history of bilateral fallopian tube ligation fifteen years back. No history of any other co-morbidities was noted. On examination, the patient's pulse rate was 82 beats per minute, blood pressure of 108/49 mm Hg, and was maintaining saturation at room air. Per-abdomen examination revealed a soft, non-distended, non-tender abdomen without any palpable mass or, visible peristalsis. There was no organomegaly. Normal bowel sounds were heard. Her routine haematological and biochemical investigations were all within normal limits except for having mild anaemia (Hb 9.2 g/dl). Other systemic examinations did not reveal anything noticeable. Abdominal radiographs taken, however, showed multiple air fluid levels with dilated bowel loops suggestive of small bowel obstruction. Contrast-enhanced computed tomography (CECT) of the whole abdomen and pelvis was done. CECT revealed features likely of midgut volvulus resulting in intestinal obstruction with long segmental ileal lipomatosis Figures .The patient was taken up for laparotomy. Intra-operative findings revealed dilated proximal bowel loops. A jejunal volvulus was noted. An approximate 45 cm length of distended ileal loop 30 cm proximal to ileocecal junction with ileal lipomatosis was noted. The mesenteric border showed relatively increased fatty deposition. There was no evidence of bowel ischemia or necrosis. The ileal segment of 45 cm containing lipomatous swellings was resected after detorsion of jejunal volvulus and hand-sewn double layer end-to--to-end ileo-ileal anastomosis was done Figures . MinimalLess than 150 occurrences of mesenteric lipomas have been documented in English literature, making them an uncommon clinical entity. Its etiopathogenetic pathway is still unclear. According to the available reports, there is a higher prevalence in individuals who are obese, have comorbidities like diabetes mellitus or hypercholesterolemia, have had trauma or radiation, have a family history of the condition, or have certain chromosomal abnormalities .Farkas et al. in their systematic review of symptomatic small bowel lipoma found that abdominal pain was the most common presentation in 68.7% as was in our case, followed by nausea and vomiting in 35.3%. Other presentations may include hematochezia/GI bleeding (33%), abdominal distension (12.2%), constipation (8.9%), and weight loss (7.5%) [Small intestinal lipomas are frequently discovered incidentally in asymptomatic people. An abdominal X-ray is useless for diagnosis. Although a mesenteric cyst may be distinguished from it by ultrasound thanks to its well-defined homogeneous echogenic mass, it is typically impacted by visceral fat or intestinal gas. Due to this, even though it is the examination of the first choice for diagnosing intestinal obstruction caused by volvulus in infants, it does not allow for the accurate definition of the tumour's boundaries or placement concerning the peritoneum . The golAs far as treatment is concerned, small lipomas are usually left untreated, however, there have been some reports of endoscopic mucosal resection of ileal lipomas , but in We report a rare case of mid-gut volvulus caused by lipomatosis of the ileum. Lipoma of the small intestine, though relatively common, very rarely causes volvulus of the intestine. The consideration of this entity as a causative factor for intestinal volvulus may help in proper treatment and prognostication, as they can be successfully managed by surgical resection.


An uncommon form of cancer known as a granulosa cell tumor (GCT) arises from ovarian sex cord cells that produce estrogen. The goal of conservative surgery in cancer is to retain organ functions while avoiding severe excision wherever possible. In oncologic gynecological surgery, fertility-sparing surgery (FSS) is a technique that tries to preserve the uterus and ovarian tissue. A 26-year-old woman with nulligravida presented with the main complaints of pain in the right iliac fossa for 10 to 15 days and fullness in the abdomen for one month, along with a change in appetite and noticeable weight loss. The Magnetic Resonance Imaging (MRI) revealed a large, well-defined, multiloculated, solid cystic mass with altered signal intensity. On exploratory laparotomy, an intraoperatively left ovarian cystic mass was seen. The ovarian mass was histopathologically diagnosed as a sex cord tumor of the ovary, with characteristics compatible with adult GCT. Disregarding the follow-up advice on discharge four months later, the patient conceived spontaneously and gave birth to a male child via emergency lower segment cesarean section. In GCTs that have not spread beyond the ovary or in people who have had relapses of the disease, FSS created the groundwork for conception and appeared safe. In the lack of any compelling supporting evidence, the line of care of terminal surgery should always be thoroughly discussed with the patient and advised for women after their families are complete.  Granulosa cell tumor (GCT) is an uncommon type of cancer that develops from estrogen-secreting ovarian sex cord cells. It compensates for more than 70% of sex cord-stromal tumors (SCST) but less than 5% of ovarian tumors. Adult GCT (AGCT) and juvenile GCT (JGCT) are two separate histological forms that exhibit various histopathological and clinical characteristics . With thIn oncology, functional and conservative surgery aims to preserve the functionality of an organ and, when possible, avoids radical resection. The goal of FSS, which is increasingly employed in oncologic gynecological surgery, is to preserve uterine and ovarian tissue . The useA 26-year-old woman who had been married for a year and was nulligravida presented with complaints of abdominal fullness for a month and pain in the right iliac fossa for 10 to 15 days. She also had a history of significant weight loss which involved 5 kg weight in six months (>5% of weight loss in six months) but had a normal appetite with no other constitutional symptoms. On examination, she was found to be obese, with a weight of 72 kg and a Body Mass Index of 32 kg/mtr2. She was pale and had a pulse rate of 100 beats/min with raised blood pressure (160/80 mmHg). The abdomen was distended up to the xiphisternum with a mass corresponding to a 36-week gravid uterus size, and it was regular and firm in consistency. However, there were no signs of lymphadenopathy or edema.The patient did not give consent for speculum examination. On per vaginal examination, fullness was present in the anterior fornix, which was cystic in consistency. The cervix deviates to the right side. Uterus size couldn’t be made due to the size and tenderness of the mass, and uterine mobility was also restricted. The ultrasonography (USG) revealed a large cystic lesion of size 23 x 27 x 26 cm with a volume of 8763 ccs with multiple internal septations within it, occupying the abdominopelvic cavity. The RMI score was 38 . The provisional diagnosis on USG was of neoplastic etiology.The patient was admitted for further evaluation of the abdominal mass. The Magnetic Resonance Imaging (MRI) revealed a large, well-defined, multiloculated, solid cystic mass with altered signal intensity, measuring 15 x 23 x 25 cm in size, suggestive of a well-defined, large, multiloculated, multiseptated lesion with the following differential diagnoses: left ovarian dysgerminoma, large abdominal echinococcal cyst, or a mesenteric lymphangioma.Her biochemical evaluation showed a reduced hemoglobin of 7.1gm/dl, and her hematocrit value was 20.90%. Her CA-125 was 38.15 units/ml and her serum lactate dehydrogenase slightly raised to 354U/L, her beta Human Chorionic Gonadotropin level was <0.10mIU/ml, and her serum inhibin A value was 7.2 pg/ml as shown in Table On exploratory laparotomy, an intraoperatively left ovarian cystic mass of 30 x 26 x 27 cm was seen with a smooth surface and intact capsule, as shown in Figures The patient had been advised to follow up with MRI and inhibin levels for up to two years and advised for using strict contraception. Injection of depot-medroxyprogesterone acetate (DMPA) was planned at follow-up visits, but she disregarded the advice on discharge, and four months later, the patient conceived spontaneously and arrived for antenatal care (ANC) and followed regular ANC visits to the hospital. She was admitted as a primigravida with a Comprehensive Gestational Age (CGA) of 37 weeks-2 days with chronic hypertension. The patient underwent an emergency lower segment cesarean section given severe preeclampsia with a poor Bishop score, and the patient was not willing to trial labor and delivered a male infant by cesarean. There was no evidence of recurrence of granulosa cell tumor at the time of delivery. She has not shown any indicators of recurrence for the past 14 months since her initial diagnosis.Different signaling routes are needed for granulosa cell proliferation, and any alteration to these signaling pathways results in excessive granulosa cell proliferation and the development of GCT. Understanding the role of these pathways in the etiology of the disease allows for the adoption of novel treatments to treat GCT, especially in the case of recurrent GCT . The FOXWang et al., in a study, concluded for young patients who want to protect their fertility, FSS is a viable alternative. In patients with an unstaged AGCT, secondary surgical staging is an important form of treatment . The sucLee et al. conducted a study on clinicopathological characteristics of GCT of the ovary and concluded that the sole component related to disease survival is the stage of the disease; therefore, FSS may be a therapy option for women with early-stage disease who want to keep their fertility . SimilarRema et al., in their study, stated that after FSS, chemo-induced gonadotoxicity compromises fertility when a patient requires chemotherapy in adjuvant. By employing GnRH analogs or oral contraceptives to downregulate hormones, the surviving ovarian tissue can be somewhat safeguarded. In their first or second decade of life, girls are most susceptible to germ-cell ovarian cancers. They typically have the early disease when they first present, making them candidates for fertility preservation. Comprehensive surgical staging also includes a unilateral salpingo-oophorectomy. Bleomycin, etoposide, and cisplatin (BEP) are used in chemotherapy to treat extra ovarian illness. Despite the fact that chemotherapy increases the chance of ovarian failure, most patients after BEP resume menstruation and have good reproductive outcomes .FSS appears to be a safe treatment option for people with GCTs and offers a possibility for women with early-stage disease who want to maintain their fertility. The degree of care for termination of surgery should always be thoroughly discussed with the patient and recommended for all women after their families are complete in the absence of any convincing supporting data.


The peripheral cemento-ossifying fibroma (PCOF) lesion primarily affects females in their second decade of living. These lesions are more frequently associated with the gingival margin, the anterior surface of the molars, and the maxilla. On clinical examination, PCOF typically appears as a well-differentiated, slowly expanding gingival mass in the interdental papilla region that is less than 2 cm in size. The surface may seem ulcerated, the base may be sessile or sometimes pedunculated, and the colour is either the same as the gingiva or reddish. The histological examination, which identifies cellular connective tissue and the focal presence of bone or calcifications, provides the basis for the final diagnosis. Treatment modalities for the PCOF include surgical excision of the lesion. A 38-year-old female reported slow-growing swelling associated with the maxillary anterior region. Removal of the lesion is done by using a scalpel, and histopathological examination revealed the peripheral type of cemento-ossifying fibroma. This case report demonstrates the management of PCOF lesions with the conventional scalpel approach with the help of proper clinical examination, radiological findings, and histopathological examination, which reveals favourable outcomes in the patient regarding esthetics and improves mastication-related issues and speech. A benign tumour called osseous fibroma mostly develops in the craniofacial bones. It comprises proliferating fibroblasts that produce osseous products, such as oval calcifications and bone, and these lesions are clearly distinguished from the surrounding bone. Ossifying fibromas can be classified as either central or peripheral. The medullary cavity enlarges in the central type, originating from the endosteum or periodontal ligament adjacent to the root apex. Only the soft tissues covering the jaws' tooth-bearing regions are affected by the peripheral type . One of PCOF is a rare lesion with variable morphologic features. It is described as a well-defined, sometimes encapsulated growth or lesion made of fibrous tissue with different concentrations of mineralised material that resembles either cementum (cementifying fibroma) or bone (ossifying fibroma) . PCOFs mA 38-year-old female with a chief complaint of a slow-growing, painless soft tissue growth between her maxillary anterior teeth reported to the outpatient department (OPD) of periodontics. Almost a year ago, the lesion began as a little papule. The patient had a history of difficulty speaking and chewing. A pedunculated, firm, non-tender growth on the papillary and marginal gingiva in connection to the maxillary right and left central incisor was found during the clinical examination shown in Figure Initially, the differential diagnoses were irritational fibroma, PCOF, pyogenic granuloma, and peripheral giant cell granuloma. As seen in Figure Measurements were taken, which revealed a growth size of 10-15 mm long and wide, as seen in Figure The patient had a history of poor oral hygiene. Considering the above findings, the patient was advised for initial therapy, surgical therapy, and haematological examination. The scaling and polishing were done before surgical treatment. Haematological analysis revealed investigation within normal range. Informed written consent was taken from the patient. Surgical intervention was carried out with the conventional scalpel technique, shown in Figure Postoperative instructions and medications were prescribed, and the patient was reviewed after seven days of surgery; on examination, the lesion was found to be healing satisfactorily as seen in Figure After 15 days of surgical intervention, a follow-up examination revealed complete wound healing and no discomfort and pain and improved esthetics at the operated site, which can be appreciated in Figure The excised specimen after the surgery was sent for histopathological examination, which confirmed the diagnosis of PCOF, as illustrated in Figure A clearly defined and sometimes enclosed lesion is called a PCOF. Fibrous tissue with varying calcific material levels that resemble cementum (cementifying fibroma) or bone (ossifying fibroma) makes up this condition. The exact cause of PCOF remains unknown, but it has been proposed that periodontal ligament cells are the source of these lesions . The occBased on the focal presence of bone or other calcifications in cellular connective tissue, a histological examination is essential for a precise diagnosis. This highlights the difficulty in diagnosing PCOF based solely on clinical observations, underscoring the requirement for the histological evaluation of biopsy specimens for a correct diagnosis . The preThis case report demonstrates that surgical excision is a simple, efficient, and affordable method for treating PCOF and giving aesthetic and functional advantages to the patient in terms of mastication and chewing. Surgical intervention and long-term follow-up results showed favourable outcomes regarding issues related to mastication and speech.


Oxidized regenerated cellulose, commonly known by the brand name Surgicel®, is a hemostatic agent widely used in various surgical procedures. While it is generally considered safe and effective, there have been reports of complications associated with its use, including the formation of pseudotumoral lesions. This article presents a case of a patient who developed a Surgicel® granuloma in the thyroid bed, mimicking a recurrent tumor. Surgicel® is known to cause a chronic inflammatory reaction, leading to foreign body giant cell formation and fibroblastic proliferation. Fine-needle aspiration (FNA) cytology is a valuable diagnostic tool for identifying pseudotumoral lesions caused by oxidized cellulose. The characteristic appearance of oxidized cellulose fragments and the presence of a granulomatous reaction can help distinguish these lesions from tumor recurrence or abscesses. To prevent Surgicel® granuloma, it is recommended to use the minimal amount necessary to achieve hemostasis. It is also important to document its use in the operative report. In cases where a recurrent mass lesion is suspected postoperatively, a comprehensive medical history, imaging studies, and FNA are essential for accurate diagnosis and management. This case report highlights the importance of considering Surgicel®-induced granuloma in the differential diagnosis of recurrent thyroid-bed tumors. A correct diagnosis can help avoid unnecessary aggressive interventions, particularly in cancer patients. Hemostasis is a crucial aspect of thyroid surgery, and various topical hemostatic agents are commonly employed as supplements to conventional methods of hemostasis . These agents have gained widespread acceptance for use in both open and minimally invasive surgeries due to their ease of application -3. In thHowever, the utilization of oxidized regenerated cellulose as a hemostatic agent has been linked to complications such as foreign body reactions and granuloma formation -9, whichIn this report, we present a case of a symptomatic neck mass that recurred two years after thyroid lobectomy, which was found to be a Surgicel® granuloma.A 42-year-old female patient was referred to the endocrine surgery clinic with a suspicious nodule in the right thyroid lobe. She had previously undergone two non-diagnostic fine-needle aspirations (FNAs). After discussing all options, the patient opted for a right thyroid lobectomy and isthmusectomy. The procedure and recovery were uneventful. The pathological evaluation of the surgical specimen revealed thyroid tissue exhibiting follicular adenoma, which was encapsulated and composed of variable-sized follicles lined by flattened to cuboidal epithelial cells with variable amounts of colloid. There was no evidence of capsular or vascular invasion. The rest of the thyroid tissue contained nodules separated by fibrovascular septa, which showed variable-sized follicles lined by cuboidal to flattened epithelial cells, and contained variable amounts of colloid. No atypia or malignancy was present. Two years after the initial presentation, the patient returned with a visible anterior neck mass underlying the well-healed surgical scar, which was causing “disfigurement and a feeling of pressure in the neck.” Ultrasound Figure  revealedAfter a thorough discussion with the patient, taking into account the symptomatic nature of the mass, the diagnostic uncertainty, and the high level of anxiety, and despite the benign pathology reported following the prior thyroidectomy, we decided to proceed with surgical excision. The tumor was primarily located in the pre-tracheal area, with adhesive extensions into the right tracheoesophageal groove, and was completely removed Figure . The surUpon reviewing the operative note from the initial surgery, it was confirmed that Surgicel® was used for hemostasis at the conclusion of the surgery.The application of hemostatic substances during surgical procedures has become a widespread practice. Among these substances, oxidized cellulose is commonly used due to its biocompatibility, bactericidal properties, and absorption characteristics ,19. SurgThe low pH also exhibits antimicrobial properties and functions as a caustic agent, facilitating blood clotting by creating an artificial clot . HoweverThe duration of oxidized cellulose dissolution depends on the amount used, with a range of two to six weeks. However, there are cases where histological evidence of oxidized cellulose fibers has been observed several years after cardiac surgery -23. AddiBesides thyroidectomy, the use of absorbable hemostatic agents, such as Surgicel®, has been investigated in other neck surgeries. For example, one study  comparedBrowder and Litwin  investigThe effectiveness of oxidized regenerated cellulose-based products, such as Surgicel®, in preventing post-surgical adhesion formation has also been investigated in several studies. While some research has indicated that Surgicel® may prevent adhesions, other studies have found inconclusive evidence .In one study, an absorbable collagen-based adhesive was found to reduce adhesions following liver injury in rats . AnotherThere have been some complications associated with the use of Surgicel® in thyroid surgery. Although studies have demonstrated no significant difference in the incidence of postoperative hypocalcemia when using Surgicel® , there hNotably, Surgicel® granulomas have been documented in various surgical procedures beyond thyroid surgery, including abdominal surgery, neurosurgery, gynecological surgery, and urological surgery -17. ThesIn experimental studies using animal models, it has been shown that oxidized cellulose can cause a chronic inflammatory reaction leading to foreign body giant cell formation and fibroblastic proliferation .Hernández-Bonilla et al.  describeThe authors evaluated 16 patients who developed these lesions after surgical procedures where oxidized cellulose was used. FNA samples were obtained from various sites, including the mediastinum, thyroid surgical bed, axilla, neck, vulva, liver, and retroperitoneum. The time interval between surgery and FNA ranged from 4 to 46 months. The cytology samples consistently showed foreign-body material with a variable granulomatous reaction. Oxidized cellulose appeared as laminated inorganic fragments with an elongated quadrangular appearance. Additional features included amorphous, ill-defined fragments and a dense proteinaceous background with phagocytic cells.FNA cytology is a valuable diagnostic tool for identifying pseudotumoral lesions caused by hemostatic agents such as oxidized cellulose. The consistent presence of foreign-body material with a granulomatous reaction and the characteristic appearance of oxidized cellulose fragments can help distinguish these lesions from tumor recurrence or abscesses.It is essential for physicians to be familiar with the clinical presentation, diagnostic approach, prevention, and management of Surgicel® granuloma. Management often involves excision due to the presence of symptoms or diagnostic uncertainty. To prevent Surgicel® granuloma, it is recommended to use the minimal amount of Surgicel® necessary to achieve hemostasis. Additionally, it is important to note the use of hemostatic agents in the operative report.This case report adds to the growing body of literature highlighting the importance of considering oxidized cellulose-induced granuloma in the differential diagnosis when evaluating patients with recurrent mass lesions postoperatively.In conclusion, Surgicel® granuloma is a possible cause of recurrent thyroid-bed tumors. To accurately diagnose and manage this condition, a comprehensive medical history, imaging studies, and FNA are crucial. This is particularly important in cancer patients, as a correct diagnosis can help avoid unnecessary aggressive interventions.


Intrahepatic cholangiocarcinoma (ICC) is frequently associated with precursor lesions, and biliary intraepithelial neoplasia (BilIN) may play a significant role in the development of ICC. However, the exact sequence and progression of these lesions remain to be elucidated. We report a rare case of ICC that exhibited extensive longitudinal intraductal extension of high-grade BilIN in the posterior bile ducts and involved the hepatic hilum and the peripheral hepatic parenchyma.A 70-year-old female presented with anorexia. Computed tomography (CT) revealed a 15 mm enhancing intrahepatic tumor extending to the right intrahepatic secondary confluence. This was associated with a 7 mm diameter cystic dilatation of the segment 6 bile duct (B6). Endoscopic retrograde cholangiopancreatography (ERCP) revealed stenosis at the bifurcation of the posterior bile duct branch. Bile cytology confirmed the diagnosis of adenocarcinoma cells. Therefore, the patient was diagnosed with an ICC involving the right glissonean pedicle and underwent a right hepatectomy and lymph node dissection. Histologic examination revealed the tumor consisted of moderately differentiated adenocarcinoma. In connection with this lesion, diffuse intraductal atypical epithelial cells, which were diagnosed as high-grade BilIN, was observed not only in the dilated B6 but in the entire posterior bile ducts, which measured approximately 120 mm in diameter. Furthermore, two distinct foci of adenocarcinomas were identified in the peripheral hepatic parenchyma. A lymph node metastasis was also present. The pathological diagnosis was ICC pT4N1M0 stage IVA. The patient underwent adjuvant chemotherapy and has shown no recurrence 5 years after surgery. Imaging modalities were unable to accurately assess the extent of the intraductal neoplastic lesions due to their low papillary or sessile intraductal tubular growth. No risk factors for BilIN development, which has the potential to predispose to cholangiocarcinoma, were identified in the present case.We present a case of ICC involving the right hepatic hilum, accompanied by extensive longitudinal extensions of high-grade BilIN and multifocal microscopic invasions in peripheral hepatic parenchyma. Notably, the intraductal lesions involved the entire posterior intrahepatic bile ducts. The presence of biliary neoplasia with extensive intraductal extension, in conjunction with ICC, should be considered as a variant of BilIN. Intrahepatic cholangiocarcinoma (ICC) is a neoplasm that originates from the endothelial cells of segmental or proximal branches of the bile duct, accounting for 5–20% of all primary liver malignancies –3. SimilA 70-year-old female presented to our hospital with complains of appetite loss and epigastric discomfort. Physical examination revealed unremarkable findings. Laboratory examination of blood showed: carbohydrate antigen 19-9 (CA19-9) level, 1369.1U/mL (> 37 U/mL); DUPAN-2, 330 U/ml > 150 U/mL); and Span-1 210 U/ml (> 30 U/mL). Liver function tests were within normal range. On computed tomography (CT), an enhancing tumor measuring 15 mm was observed, which extended to the right secondary biliary confluence Fig. . Based o0 U/mL; aWe present a rare case of ICC that is associated with extensive longitudinal intraductal extension of high-grade BilIN. This case is unique, because the epithelial tubular neoplasm, characterized by severe nuclear atypia, exhibited diffuse and continuous extension throughout the entire bile duct of the posterior segment. In addition, this lesion secondarily involved in the perihilar hepatic parenchyma. Progressive stages of this condition are also associated with two foci of minimally invasions in the peripheral hepatic parenchyma. In this case, the tumor exhibited perineural and lymphovascular invasions, and one regional lymph node metastasis was observed.Anatomically, ICCs are categorized as small duct ICC or large duct ICC –6, 10. LBilIN has been reported as a preneoplastic condition that has the potential to progress to invasive cholangiocarcinoma –9. PathoImmunohistochemical results of the BilIN lesions showed negative expression of MUC1 and MUC2, but positive expression of MUC5AC ( +). In contrast, the perihilar tumor exhibited positive for MUC1 and MUC5AC, but negative for MUC2. Zen Y et al. reported different expression patterns of MUCs and cytokeratins in neoplastic biliary epithelia of BilIN and IPNB during progression to ICC . FrequenLymph node metastasis (LNM) has been one of the strongest predictors of poor outcome among patients undergoing curative-intent resection for cholangiocarcinoma –22. ZhanSurgical margins should also be carefully evaluated and taken into consideration during clinical procedures. In a recent meta-analysis investigating the effect of surgical margin width on OS in patients with ICC, it was found that negative surgical margins are associated with improved OS and DFS following surgical resection . SpecifiWe present a case of ICC involving the right hepatic hilum, accompanied by extensive longitudinal high-grade BilIN lesions and multifocal microscopic invasions in peripheral hepatic parenchyma. The presence of biliary neoplasia with such extensive intraductal extension, in conjunction with ICC, should be considered as a variant of BilIN.


The Herlyn-Werner-Wunderlich syndrome (HWWS) is characterized by the triad ofuterus didelphys, obstructed hemivagina, and renal agenesis. The typicalclinical presentation involves chronic pelvic pain, dysmenorrhea, and palpableabdominal mass, related to hematocolpos/hematometra. It is a rare disease, witha challenging clinical and radiological diagnosis. Surgery is the definitivetreatment. Complications such as endometriosis, infertility and chronic pelvicpain occur more frequently and severely when diagnosis and treatment aredelayed. This is a case report of a twelve-year-old patient admitted to theGynecology Department of the Federal University of Rio de Janeiro’s GeneralHospital (HUCFF/UFRJ), in March 2021, with progressive symptoms of dysmenorrheaand abdominal distention due to palpable abdominal mass. She had a previoushistory of congenital solitary kidney. Magnetic Resonance Imaging (MRI) showed adouble uterus with hematometra and hematocolpos on the left side, pelvicendometriosis and left renal agenesis. Conservative clinical treatment withinhibition of the hypothalamic-pituitary-ovarian (H-P-O) axis was initiatedwhile a definitive surgical approach was being defined. In June 2022, thepatient underwent left hemi-hysterectomy and salpingectomy, achieving fullremission of symptoms. Given the rarity of this syndrome and its potentialcomplications, our report aims to familiarize clinicians with it, mostly thosewho work with children and adolescents, so that more patients have access toearly diagnosis and adequate treatment. Consequently, future fertility can beeffectively preserved. Mullerian anomalies are a rare group of anatomical malformations of the femalegenital tract. They occur due to alterations during the development and fusion ofthe Müllerian ducts (or paramesonephric ducts), which normally develop intothe uterus, uterine cervix and the two upper thirds of the vagina. Mulleriananomalies are commonly followed by alterations in the development of the Wolff ducts(mesonephric), which later originate the kidneys, justifying the frequentassociation between Mullerian malformations and urinary tract disorders  is a rare congenital malformation of thefemale urogenital tract composed of the triad: uterus didelphys, obstructedhemivagina and renal agenesis. Its incidence ranges from 0.1-3.8% in the femalepopulation  in March 2021 withsymptoms of abdominal pain beginning right after menarche, associated with aprogressive abdominal distension. In her early childhood she was diagnosed with asolitary right kidney, properly located. She was diagnosed with theHerlyn-Werner-Wunderlich syndrome and was submitted to a left hemi-hysterectomy withipsilateral salpingectomy in June 2022, evolving with complete remission ofsymptoms.Our main goal is to reinforce the importance of acknowledging Mullerian malformationsin order to maintain an appropriately high index of suspicion, enabling earlydiagnosis and adequate treatment. Therefore, we are able to improve our patient’squality of life and reduce the frequency and severity of complications such asinfertility.This is the case of a 12-year-old patient, brown skinned, born in Rio de Janeiro,Brazil, attending middle school, without previous sexual activity, with a history ofmoderate to intense dysmenorrhea, which started right after menarche, refractory tocommon analgesics, associated with progressive increase in abdominal volume. InMarch 2021, she underwent an abdominal pelvic ultrasonography, which revealed alarge complex image, possibly originating from the left ovary. For this reason, shewas referred to a quaternary care unit and then admitted to our Clinic at theHUCFF/UFRJ.The patient had a previous diagnosis of congenital - and anatomically positioned -solitary right kidney, found in a scintigraphy performed at one year of age(March/2010). There were no other relevant morbid or family/social antecedents. Shehad thelarche and pubarche at age nine, followed by menarche at eleven. Her fivefirst menstrual cycles were regular, with normal duration and flow and after that,progressive dysmenorrhea ensued.The physical examination revealed adequate Tanner stage (M3P3) and a palpable massoccupying the hypogastrium and left iliac fossa, measuring approximately 10 cm,mobile and painful. Vulvar inspection and speculum exam were normal. Gynecologicbimanual (two-handed) palpation of the uterine corpus found a mass bulging thecul-de-sac on the left side and collapsing the vagina.A new pelvic US performed at our institution again in March 2021 showed an elongatedimage with heterogeneous content with debris (10.7x7.6x6.5cm), compatible with bloodcontent, located in the left iliac fossa, laterally adjacent with the right uterinehorn. The possibility of left uterine horn with hematometra was considered and thehypothesis of congenital anomaly of the genital tract with consequent obstructiveuterovaginal syndrome was raised. We decided to proceed the investigation with apelvic MRI for diagnostic confirmation and elaboration of a therapeutic approach. Inparallel, medroxyprogesterone acetate 150 mg (injectable suspension) was started inorder to control signs and symptoms. The patient was well adapted to the method andevolved with secondary amenorrhea. However, she occasionally had episodes ofworsening pain, seeking emergency medical care for parenteral analgesia.A pelvic MRI was performed in May 2021, demonstrating uterine duplicity withsignificant hematometra and hematocolpos on the left side , 1B, 1C Considering the MRI and hysteroscopic findings, a surgical approach was planned,initially via vaginoscopy, with the aim to drain the left hematocolpos andhematometra. If vaginal access was not feasible, a laparotomy approach was suggestedat the same surgical time for left hemi hysterectomy. The surgical plan waspresented to the patient and her guardians, who understood and approved it, signingan informed consent form.The patient underwent surgery in July 2022. After an unsuccessful attempt atvaginoscopy under sedation, laparotomy was performed with left hemi hysterotomyfollowed by drainage of hematometra and hematocolpos . PosteriThe Herlyn-Werner-Wunderlich syndrome (HWWS) is a rare mullerian anomaly,characterized by the triad: uterus didelphys, obstructed hemivagina and ipsilateralrenal agenesis. It was first described in 1922  and Müller (paramesonephric) ducts  or septal resorption failure proposed a subclassification of HWWS into type 1 and type 2, according to the typeof obstruction . The preferred approach is laparoscopic, as it isless invasive, but laparotomy can be used if laparoscopy is not available, withacceptable results . It was therefore concluded thatlaparoscopy should only be performed in patients, refractory to medical treatmentand with evidence of deep/invasive disease (The Herlyn-Werner-Wunderlich syndrome should be known by clinicians who are involvedwith child and youth health care in order to improve these patients’ quality oflife. The diagnosis is clinical and radiological, and should be confirmed as earlyas possible in order to reduce further complications. Future fertility is one of themain concerns, which can be successfully preserved, even though obstetriccomplications occur more often. The definitive treatment is surgical, although thebest timing is still under debate. The approach must be individualized according tothe anatomical variation presented by the patient. A multidisciplinary approachinvolving clinicians, surgeons and radiologists is essential to achieve the bestoutcomes.


Choriocarcinoma is a highly aggressive malignant tumor that occurs due to the formation of an abnormal trophoblast. Choriocarcinoma is classified into gestational (GC) and nongestational (NGC) subtypes. The majority of nongestational diseases are limited to ovaries. Extragonadal NGC is a sporadic occurrence and a diagnostic and therapeutic dilemma. Here, we present a young 24-year-old female who presented with a widespread metastatic disease to the brain, bilateral kidneys, lungs, liver, pancreas, and small bowel. She was diagnosed with extragonadal NGC, probably originating from her kidneys. She responded poorly to standard first-, second-, and third-line chemotherapies. Detailed literature analysis with various aspects of pathogenesis, diagnostic criteria, clinical presentation, and treatment options are discussed. There is an unmet need for further research and consensus on many aspects of this rare disease. Choriocarcinoma is a highly aggressive malignant tumor that occurs due to the formation of an abnormal trophoblast. It consists of two types of cells: cytotrophoblasts and syncytiotrophoblasts. The cytotrophoblasts are primitive mononuclear trophoblastic stem cells. The syncytiotrophoblasts are multinucleated cells formed by the fusion of underlying cytotrophoblasts and are the differentiated hormone-secreting components that secrete beta human chorionic gonadotropin hormone (beta-hCG) . ChoriocGC is a rare pregnancy complication with an incidence of one in 20,000 to one in 25,000 in Western countries. Most cases of GC are intrauterine. Extrauterine GCs may also originate at a site of ectopic pregnancy in the ovary, fallopian tube, abdominopelvic cavity, or other sites. The estimated incidence of gestational ovarian choriocarcinoma is one in 369 million pregnancies. The gestational type is usually preceded by a molar pregnancy, term pregnancy, ectopic pregnancy, or abortion within one year of the antecedent event .NGCs are sporadic neoplasms amounting to less than 1% of all choriocarcinomas. NGCs develop from pluripotent germ cells, most commonly in the gonads. Gonadal NGC amounts to 0.6% or less of all ovarian tumors, making it one of the rarest ovarian tumors ,3. OvariExtragonadal NGCs are even more uncommon; are located in midline organs, such as the mediastinum, retroperitoneum, and pineal gland; and are called germ cell tumors. These are primarily reported in males and are usually associated with poorly differentiated somatic carcinomatous components . ExtragoThe microscopic appearance of GC and NGC are similar, leading to delays in the diagnosis of NGC and often underdiagnosis or misdiagnosis in clinical practice ,2,3. TheHere, we report a rare extragonadal widespread metastatic NGC, probably of renal origin, in a 24-year-old lady with clinical and histopathological findings refractory to standard chemotherapy.2P2L1A0D0) with an Eastern Cooperative Oncology Group (ECOG) performance status of one, was referred to our hospital for management of brain lesion, which was operated twice outside. Her last childbirth was 18 months before the start of symptoms, and she denied any abortion or ectopic pregnancy after her previous childbirth. She initially presented to the neurosurgery department with constant headaches, generalized seizures, and left-sided weakness. Her magnetic resonance imaging (MRI) brain was done, which was suggestive of multiple (three in number) well-circumscribed heterogeneous lesions with mildly increased diffusion signal, the intralesional subacute hemorrhagic signal in the right occipital lobe, high frontoparietal lobe, and left parietal lobe. A significant edema surrounded the lesions with a midline shift of 5 mm to the left side. The most significant lesion was 25 x 21 mm, likely neoplastic lesions  brain was suggestive of no midline shift, and the lesion in the right high frontoparietal lobe was not visualized as post-operative changes replaced it. Histopathology was compatible with metastatic, poorly differentiated carcinoma. After a month, she had another seizure episode and was reviewed by the neurosurgery team. Her repeat MRI brain revealed additional well-circumscribed heterogeneous lesions with mildly increased diffusion signal intralesional subacute hemorrhagic signal in the right occipital lobe and left parietal lobe. The most significant lesion measured 48 x 35 mm and was associated with edema and a midline shift of 20 mm, noted to the right side. She underwent left occipital and right occipital craniotomy and micro-surgical tumor decompression.The patient was referred to our center for further management. Whole-body positron emission tomography and computed tomography (PET-CT) were suggestive of hypermetabolic nodules in the right lung lower lobe with other non-fluorodeoxyglucose (FDG) avid nodules likely neoplastic and minimal right pleural effusion. The right kidney showed a lesion involving the lower and interpolar cortex, approximately 4.8 x 4.5 x 5.9 cm in size, with a maximum standardized uptake value (SUV-max) of 14.2. The left kidney showed a mass lesion involving the interpolar cortex, measuring approximately 2.9 x 2.6 x 3.5 cm, with an SUV-max of 9.7. There was evidence of craniotomy with an ill-defined lesion in the parietal lobe, and an uptake is seen in the jejunal bowel loops (SUV-max 9.3). FDG avid lesions, the largest of size 1.3 x 1.1 cm, were noted in segments VIII, II, and IV of the liver. There was focal uptake noted in the tail of the pancreas, with an SUV-max of 5.0 and subtle centimeter-sized hypodensity with an SUV-max of 12.1 in the jejunal loop Figure , C, D, EHer histopathology review of brain tumor tissue suggested a high-grade neoplasm with a differential of mixed germ cell tumor Figure . ImmunohThe case was discussed in our institutional multidisciplinary board and was planned for whole-brain radiotherapy (WBRT) and palliative chemotherapy. The patient was given WBRT in 30 Gy/10 fractions. The patient was then started on multiagent chemotherapy with a standard EMA-CO  regime. The patient defaulted for more than two weeks after the first cycle of EMA-CO, leading to a surge in beta-hCG values to 55,027 mIU/ml before the next dose. A total of seven cycles (EMA-CO) were given with the monitoring of beta-hCG levels. The beta-hCG values never normalized, and a plateau was seen after the sixth cycle  regime, which she has received five cycles to date and tolerated well. Her recent beta-hCG level is 73.2 mIU/ml, and currently, she is asymptomatic.Due to the sporadic occurrence of NGC, its clinical characteristics are unclear, and the presenting symptoms are usually nonspecific. They may present with precocious puberty in young girls. Depending upon the disease site, they can have symptoms, such as abdominal pain, abnormal uterine bleeding, pelvic lump, cough with hemoptysis, hematuria, seizures, and vision abnormalities -5. Our cHistorically, Saito's criteria were postulated in 1963 as diagnostic criteria for NGC [Thirty-nine cases of ovarian NGC were retrospectively analyzed by Liu et al. in 2020 . Sixty-eSome studies propose that a prior pregnancy history should rule out a diagnosis of NGC -8. A choBecause of its sporadic occurrence, the staging of NGC remains a dilemma. Since ovarian or gonadal NGC is a specific type of a ovarian germ cell tumor, staging for ovarian cancer is applied in most reported cases ,3. ThereSome studies have shown that NGC is less chemo-sensitive and has a poorer outcome than GC ,3,4. EMAThe site of origin of our case cannot be commented on with certainty, given the widespread metastasis in the brain, lungs, liver, pancreas, bilateral kidneys, and small intestine. Metastases of NGC to the kidney are rare, and bilateral metastasis is even more scarce. In our case, the most prominent lesions were noted in bilateral kidneys and can be assumed to be the primary site of origin. We could not do genetic analysis due to resource constraints for ascertaining the origin. Our diagnosis of NGC with a probable kidney origin was also based on history, histopathology, imaging, clinical presentation, and tumor behavior. The patient has responded poorly to standard regimes, such as EMA-CO, EMA-EP, TP/TE, and GEMCAP. She never had a sustained reduction of beta-hCG values, again proving the aggressive nature and poorer response of NGC compared to GC.Next-generation sequencing (NGS) is being used for detecting various targets, which could be prognostic or predictive and may guide the development of novel therapies. Different targets, such as STK11, SMARCA4, and EGFR V774M, have been discussed by Ma et al.  and OnisThere are many unmet needs or dilemmas in diagnosing and managing NGC. The preferred staging system to be applied is ovarian, or GC is unclear. No defined prognostic model exists, and the WHO GC model cannot be applied. Saito's criteria are ancient and probably need revision. The revised criteria were used by Shao et al. , but theDiagnosing NGC is a clinical challenge, citing a lack of proper criteria and vague clinical presentation. The role of obstetric and menstrual history is vital in diagnosis. This case highlights the vague presentation leading to delay in diagnosis and suboptimal response to standard chemotherapy options. This case has summarized the existing literature on NGC and is likely to help clinicians diagnose and manage future similar cases. Many unmet needs and dilemmas are summarized and brought to readers' attention. In the context of this case discussion, the need for further research in this area and consensus regarding diagnostic criteria and treatment options, among others, are reinforced.


Langerhans cell histiocytosis (LCH) is a rare disorder. The treatment options vary depending on how many organs are involved and how extensive the disease is. In this report, a case of LCH with isolated 6th cervical vertebra (C6) collapse was presented. This case was treated with anterior corpectomy and instrumented fusion, followed by local radiotherapy (RT), with a good clinical outcome up to postoperative six months.This was a 47-year-old female patient with a complaint of neck pain and bilateral shoulder pain for two months before consultation. She was initially treated with analgesics, but the pain was persistent. Further radiological evaluations revealed an osteolytic lesion within the C6 vertebral body with a pathological fracture. Magnetic resonance imaging (MRI) with contrast of the cervical spine revealed diffused hypointense signal changes on the T1-weighted images and hyperintense signal changes on the T2-weighted images in the C6 vertebral body, with significant contrast-enhanced infiltration signals. Furthermore, in positron emission tomography-computed tomography (PET-CT), focal hypermetabolism and abnormal uptake signals were seen only in the C6 vertebral body. The patient underwent an anterior cervical corpectomy with instrumented fusion. The histopathological results confirmed the diagnosis of LCH. The patient reported significant pain relief on postoperative day one. Moreover, she was treated by local RT at postoperative one month. Good clinical outcomes were achieved in the form of no pain and recovery in neck mobility up to postoperative six months. No evidence of recurrence was observed at the final follow-up.This case report describes a treatment option for a solitary C6 collapse with LCH managed by anterior corpectomy and instrumented fusion, followed by local RT, with a good clinical outcome at postoperative six months. More studies are needed to elucidate whether such a treatment strategy is superior to surgery or RT alone.The online version contains supplementary material available at 10.1186/s12891-023-06543-2. Langerhans cell histiocytosis (LCH) is a rare neoplasm of hematopoietic myeloid precursor cells that tends to affect Caucasian male children, with a peak incidence around the age of one to three . LCH canThis was a 47-year-old female patient who presented with continuous pain in her neck area and the back of her bilateral shoulders for two months before she came to our hospital for consultation. Her past medical history was healthy, except that a pulmonary nodule had occasionally been seen on a low-dose chest computed tomography (CT) scan. Observation was advised on the nodule, as there were no signs of malignancy. She did not have any family history of malignant tumors or any injury before the onset of neck pain. In addition, she did not have any episodes of fever, significant loss of body weight, or poor appetite after the onset of the pain. The neck pain was mechanical in nature. There was no radiating pain to bilateral upper limbs or numbness over the upper and lower limbs. She was treated in the form of nonsteroidal anti-inflammatory drugs and physical therapy, but the pain persisted and increased in severity over time. On physical examination, a significant reduction in the range of motion of her cervical spine was noted: forward flexion 20 degrees, extension 20 degrees, left and right side bending 25 degrees, and left and right rotation 30 degrees, with marked spasms and tenderness over the paravertebral area. There were no neurological deficits in the upper or lower limbs.For the laboratory data, the complete blood count test, tumor markers, C-reactive protein, and erythrocyte sedimentation rate were all within normal levels. The cervical spine plain radiography revealed that there was collapse in the C6 vertebral body, with mild malalignment in the lateral view. A pathological fracture was considered Fig. . The conOpen surgery in the form of a C6 corpectomy with instrumented fusion was performed. The procedure included removing nearly the entire vertebral body (corpectomy) of C6 together with the adjacent discs (discectomy). The resulting bone void after corpectomy and discectomy was filled with bone graft for fusion and stabilized by instrumentation, which included the use of a titanium cage, a plate, and four screws of an appropriate size. Intraoperatively, discectomies of C5-6 and C6-7 were performed. Then, the C6 vertebral body was excised in a subtotal manner. It was observed that the bone lesion involved part of the posterior wall of the vertebral body and part of the PLL. Furthermore, adhesion between the PLL and the dural membrane was noted. The excised tissue was then sent for a frozen section, routine pathology, and tissue culture for microbes. The intraoperative report of the frozen section showed scanty sheets of uniform ovoid cells with eosinophilic cytoplasm. No distinct pleomorphism, mitosis, or necrosis were noted. Such findings favored neoplastic lesions. However, it could not be concluded from the frozen section whether the condition was benign or malignant. Thus, intraoperatively, the decision was made to proceed with an intralesional resection, and the tumor-like tissues were removed as much as possible. A titanium mesh cage filled with granular cancellous bone allograft was inserted between the C5 and C7 vertebral bodies for fusion  at postoperative one month, as advised by the oncologist. The patient was followed up at postoperative two months and six months, and showed good clinical outcomes with no pain in the neck and shoulder and a significantly improved range of motion of the cervical spine at last follow-up: forward flexion 40 degrees, extension 35 degrees, left and right side bending 45 degrees, and left and right rotation 60 degrees. Cervical spine plain radiography was repeated Fig. . The cagLCH was first described by Paul Langerhans Jr. in 1869 . LCH refLCH is a rare disease that usually affects children. The age-adjusted incidence rates 0–14 years) were reported to vary between 2.6 , 5 10],,10], and4 years wPain is a very frequently presented symptom in LCH patients , 20. HuaPlain radiography is important for diagnosing and staging LCH . HoweverFor adults with LCH, there are no standard therapies, and prospective trials have not been performed . BecauseIn a report by Vielgut et al. , a similThe use of RT in LCH was reported as early as 1979 , in whicIn summary, the case of a 47-year-old female with a solitary LCH lesion in C6 with vertebra collapse is reported. Our case differs from the representative cases reported in the literature, in which LCH is generally involved in smaller areas of the vertebral bodies in adults, and vertebral collapse is less common –16. The LCH is a rare disorder, and it can present in heterogeneous ways. Treatment options vary depending on how many organs are involved and how extensive the disease is.Surgical excision and instrumented stabilization are considered if a solitary lesion leads to bone destruction and instability of the skeleton system .Postoperative radiotherapy is an option intended for better local control of recurrence. However, no evidence has shown the superiority of a strategy in which surgery followed by RT is better than either surgery alone or local RT alone for the treatment of solitary LCH bone lesions.Additional file 1.


Endometrial stromal sarcoma (ESS) is a rare disease in patients with uterine malignancies, accounting for <1%. Low-grade endometrial stromal sarcoma (LGESS) accounts merely 0.2% of gynecologic malignant tumor. Primary low-grade extrauterine endometrioid stromal sarcomas (LGEESS) is even more uncommon, with only a few documented case reports. We report a case of primary LGEESS exhibiting widely invasion in multiple organs after hysterectomy, which is the first case reported in Jiangsu Province of China.A 42-year-old nulliparous female with dysgnosia presented with a moderate amount of irregular vaginal bleeding, abdominal pain and distension, and frequent urination for 2 days. Her surgical history included a total hysterectomy and bilateral salpingectomy for uterine fibroids 6 years ago. Ultrasonography and the abdominal and pelvic computed tomography scan detected some solid polycystic masses in the pelvic and abdominal cavities.The histopathology of the specimen confirmed the diagnosis of LESS in the absence of florid endometriosis. The patient was diagnosed with primary extrauterine endometrial stromal sarcoma at FIGO stage III.Surgery and histopathology were performed.After surgery, the patient was maintained on leuprorelin acetate microspheres with sustained release for injection at 3.75 mg once every 4 weeks while refusing further radiotherapy.The diagnosis of primary LGEESS is challenging mainly because of their unforeseen location and nongynecologic signs and symptoms. Total hysterectomy and bilateral salpingo-oophorectomy are recommended to LGESS, while additional resection for extrauterine disease depends on disease extent and resectability. LGESS accounts for merely 0.2% of the gynecologic malignant tumor. It is even more uncommon for primary low-grade extrauterine endometrioid stromal sarcomas (LGEESS) because it develops at extrauterine sites especially including the peritoneal surfaces, intestinal wall, ovaries, pelvis, vagina, urinary bladder, and fallopian tubes, without involving uterine.,3Endometrial stromal sarcoma (ESS) is a rare disease in patients with uterine malignancies accounting for <1%. 2020 WHO classification schema has identified ESS as low-grade endometrial stromal sarcoma (LGESS) and high-grade ESS.Here, we report a case of primary LGEESS exhibiting wide invasion in multiple organs after hysterectomy, which is the first case reported in Jiangsu Province of China.A 42-year-old nulliparous female with a diagnosis presented with a moderate amount of irregular vaginal bleeding, abdominal pain and distension, and frequent urination for 2 days. Her surgical history included a total hysterectomy and bilateral salpingectomy for uterine fibroids 6 years ago. Per vaginal examination revealed a 15-mm-diameter red lesion on the vaginal stump. On gynecological examination, a 10-cm-diameter nonmobile lump was palpable with tenderness over the right adnexa.Ultrasonography detected some solid polycystic masses in the pelvic and abdominal cavities  . However, when they do, they pose challenges in diagnosis. Extra-uterine manifestation and histological features, in conjunction with immunohistochemical and molecular findings, are essential for making an accurate diagnosis. Although extremely rare, treatment guidelines of extra-uterine LGESS are based on those for uterine ESS. Advance trials with a larger number of patients, longer follow-up times, and uniform treatment modalities are needed to clarify the roles of chemotherapy, hormonal therapy, and radiotherapy in LGESS.Investigation: Ningxin Chen.Project administration: Min Gong, Qiaoling Liu.Resources: Qiaoling Liu.Supervision: Min Gong, Qiaoling Liu.Writing – original draft: Ningxin Chen, Min Gong, Wen Lai.Writing – review & editing: Ling Ling, Qiaoling Liu.


A 23‐year‐old male with a tumor in the eye socket was characterized by multimodal images, including ultrasonography, computed tomography, and magnetic resonance imaging. After admission, surgical resection of the tumor was performed and superficial angiomyxoma was confirmed. Two years later, this tumor recurred in the same location.Superficial angiomyxoma (SAM) is a rare benign neoplasm composed mostly of myxoid material that can affect many parts of the body in middle‐aged patients. Only a few case reports have involved imaging, which is extremely insufficient. Here, we present a case of SAM in the eye socket evaluated by imaging, including ultrasonography, computed tomography, and magnetic resonance imaging. The patient underwent surgical resection, and the diagnosis of SAM was confirmed. During the postoperative follow‐up, the tumor recurred in the same location without metastasis 2 years later. Superficial angiomyxoma is an uncommon, benign, multilobular neoplasm occurring mostly in middle‐aged patients. Here, we presented a case in the eye socket of a 23‐year‐old male characterized by ultrasonography, computed tomography, and magnetic resonance imaging as well as confirmed with pathology. Superficial angiomyxoma (SAM) is an uncommon, benign, multilobular neoplasm composed mostly of myxoid material that shows sparse cellularity and abundant vascularization. According to the previous literature, solitary SAMs are frequent on the head or distal areas of the limbs, which are not associated with the Carney complex; multiple SAMs in patients with the Carney complex are usually located on the eyelids and in the external auditory canal.A 23‐year‐old male was admitted to our hospital for the evaluation of a tumor in his right eye socket, which was present for more than 2 years. After admission, an ophthalmological examination revealed that the tumor was tough, immovable, and smooth and caused no pain. Initially, ultrasonography of the eyes revealed the tumor was located in the area of the inferior lacrimal sac Figure . Next, cSuperficial angiomyxoma is an extremely rare benign tumor occurring mostly in middle‐aged patients; both males and females are equally affected. This tumor can affect many parts of the body, although the head, neck, trunk, and limbs are the most likely locations. Palpation of SAM reveals a soft, mobile, nonulcerated, and well‐defined tumor, with a range of polypoid to papulonodular appearances. On ultrasonography, SAM is a hypoechoic and heterogeneous tumor with a regular shape, well‐defined boundary, capsule, and posterior acoustic enhancement, which has a great variety of blood flow signals.Due to the rarity of SAM, it is uncommon for surgeons to accurately predict, or misdiagnose it as a common benign soft tissue tumor, and biopsies or simple excisions are normally carried out.Guiwu Chen: Data curation; writing – original draft; writing – review and editing. Wenqin Liu: Data curation; writing – original draft; writing – review and editing. Yongjie Cao: Investigation; validation; visualization. Xiaodong Guo: Data curation; validation; visualization. Yuhuan Xie: Project administration; supervision; writing – review and editing.No funding was received for this study.The authors declare no conflicts of interest.We can confirm that this manuscript is an honest, accurate, and transparent account of the case being reported and that no important aspects of the case have been omitted.The corresponding author had the written consent of the patient to use the data for publication.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.