Basidiobolomycosis is a rare fungal infection caused by saprophyte Basidiobolus ranarum. It is rarely seen in healthy adult patients; however, it usually affects children. The commonly involved sites are skin and subcutaneous tissue, mostly found in the Middle East and the southwestern United States. The diagnosis is challenging because of the lack of specific clinical presentation and the absence of predisposing factors.In our case report, we discuss a 38-year-old male patient who presented with a 2-months history of right lower quadrant pain. Initially, his pain was intermittent and gradually increased in intensity; it localized to the right lower quadrant and radiated to the right flank region. No relieving or aggravating factors were noted. In addition, the patient mentioned a history of constipation, weight loss, decreased appetite, and vomiting-however, no history of fever, night sweats, trauma, or recent travel. The diagnosis was made based on computerized tomography (CT) guided biopsy of the mass, illustrating the findings of fungal hyphae with a gradual increase in the eosinophilic count since admission. The patient was managed using a combined medical and surgical approach, including surgical debulking of the mass and a well-monitored course of anti-fungal therapy.Gastrointestinal basidiobolomycosis infection (GBI) can present in many forms, with an increasing potential to invade the colon, ultimately forming an inflamed mass. Nonetheless, the presence of a mass invading the colon, adjacent vessels, and a retroperitoneal area, along with an increase in the number of eosinophil count in the Middle East region, should raise the suspicion of basidiobolomycosis fungal infection. Basidiobolomycosis is an unusual fungal infection caused by Basidiobolus ranarum . This saWe report a case of GBI in a middle-aged male invading the right colon, adjacent vessels, and retroperitoneal area. The case is unique both in clinical presentation as well as in disease distribution involving multiple organs. GBI is usually confined to a single organ, but it was seen infiltrating retroperitoneal space and encasing the superior mesenteric vessels in this case. The treatment plan for the current case was based on a combination of surgical resection and intravenous anti-fungal medications.A previously healthy 38-year-old male presented in our emergency department at Hamad General Hospital with progressive colicky abdominal pain for two months. The pain was intermittent, localized to the right lower quadrant, and increased in intensity a few days before admission. The pain radiated to the right flank and was not associated with any aggravating or relieving factors. Other symptoms in his history included weight loss, constipation, loss of appetite, and vomiting, but no obstructive symptoms were noted. There was no history of fever, night sweats, trauma, or unusual habits. He had no medical history nor any family history of colon cancer.He was alert, oriented, and not in distress on physical examination. His abdominal examination revealed a palpable mass in the right iliac fossa, which was mildly tender and fixed. In addition, no signs of peritonitis or distension were noted. His digital rectal examination was normal. His initial laboratory results were as follows Table .A computerized tomography (CT) scan of the abdomen Figure  revealedThe patient was then admitted to the hospital and was started on empirical antibiotics alongside a bowel rest regime. In anticipation of his prolonged fasting state and undernourished condition, the patient was considered the right candidate for Total Parenteral Nutrition (TPN) to optimize his nutritional status. The patient's clinical status remained stable while being treated conservatively; therefore, a colonoscopy Figure  was arra3/ul). The mass eventually became tender and larger, leading to multiple spikes of high-grade fever, GI disturbance in the form of rectorrhagia, and frank hematuria. This resulted in a significant hemoglobin drop that required multiple blood transfusions. Due to the uncertainty over his diagnosis, it was decided to proceed with Magnetic resonant enteroclysis (MRE) to delineate the mass further . The radiological evidence of involvement of other structures, namely the ureter and bowel, along with a rapid increase in the size of the mass raised the suspicion of an aggressive malignant condition, hence, the reluctance to offer a non-curative surgery without establishing a diagnosis. To reach a diagnosis, a CT-guided biopsy of the mass was performed.It revealed a partly necrotic fibroconnective tissue containing scattered fungal organisms with associated multinucleated giant cells with a background of numerous eosinophils. The morphological features were compatible with basidiobolomycosis. The infectious disease team was involved, and the patient was immediately started on empiric anti-fungal treatment . Despite being on aggressive medical treatment, his condition did not improve. On the contrary, he became more septic. He started to show signs of deterioration in the form of tachypnoea 25-28 (breaths /min) and tachycardia 130-135 beats /min with a temperature reaching above 39 degrees Celsius.He was resuscitated and taken to the operating room for exploratory laparotomy. During the surgery, we found a huge mass involving the root of the small bowel mesentery, distal to the superior mesenteric artery (SMA) origin. The mass also involved the distal ileum, cecum, and ascending colon up to the hepatic flexure. The mass was also seen extending into the retroperitoneum on the right side. Part of the mass contained ischemic ileal loops with no obvious perforation Figure . We did Damage control surgery was performed, and his abdomen was left open. The total operative time was 3 hours, and the intraoperative blood loss was about one liter. Forty-eight hours post-surgery, he was taken back to the operating theatre for a second-look laparotomy. During the second exploration, we thoroughly examined and ran the bowel, which appeared healthy; therefore, the end of the small bowel was brought out as end ileostomy, and the abdomen was closed. Post-surgery, the patient was kept in the surgical ICU for a week. During his stay in the critical unit, he showed gradual recovery and was transferred to the floor on postoperative day seven. he was started on anti-fungal (voriconazole IV). He had a slow recovery and was sent home in good condition after being kept on the ward for two weeks. His last follow-up ileostomy is planned to be closed after completing his oral terconazole, which was prescribed by the infectious disease team. Gross examination of the main resected specimen revealed a predominantly necrotic mass involving the large bowel wall, widely extending into the surrounding mesentery. Histopathology showed widespread zones of necrosis, interspersed with areas of fibrosis and heavy eosinophilic infiltrate and areas suggestive of the splendor-hoeppli phenomenon. Amongst this extensive inflammatory reactions were numerous variable-sized thin-walled and broad hyphae, diagnostic of basidiobolomycosis. The fungal wall staining was intensified by Periodic Acid Schiff (PAS) and Grocott's figure -7.Visceral basidiobolomycosis is a fungal infection of visceral and gastrointestinal tissues. The first case of GIB was reported in a Nigerian child in 1964 . RegardiThe unusual presentation in an adult male makes the diagnosis of such entity challenging, as published evidence supports such cases to be common among children. In terms of diagnosis, signs, and symptoms of GBI are largely non-specific during clinical presentation . Indeed,Concerning laboratory findings, our patient blood tests revealed elevated ESR, eosinophilia, and neutrophilic leucocytosis. All laboratory findings in our patient were consistent with gastrointestinal basidiobolomycosis . As mentThe difficulty in preoperative diagnosis mentioned in several cases in the literature suggests the importance of preoperative diagnostic procedures that should be used in such cases as colonoscopy and biopsy; however, establishing the diagnosis was difficult (8). In our case, CT-guided biopsy was the most useful tool in narrowing the differentials, thus leading to a final diagnosis. On the other hand, the biopsy taken from the colonoscopy was unsatisfactory and misleading.Treatment of this rare fungal infection still lacks clear consensus from experts. Although combination therapy with surgical excision and anti-fungal has been historically used, a few published reports have described clinical improvement with anti-fungal therapy alone ,11. TherThis aggressive fungal infection is difficult to diagnose and treat and continues to impose a challenge for physicians and surgeons. Our case report reveals that early recognition of the disease and commencement of the right treatment can result in good clinical outcomes. Perseverance with medical treatment in our case was futile and resulted in deterioration of the patient's condition forcing emergency laparotomy. Therefore, we conclude that in such conditions, once a diagnosis is established, the treatment is primarily surgical.


Acute appendicitis is a highly common cause of abdominal pain that presents with nausea and vomiting, characteristic physical exam findings, and imaging evidence of appendiceal inflammation. In the absence of these signs, diagnosis can be difficult. This case report demonstrates an uncommon presentation of appendicitis that led to a delay in diagnosis and aims to discuss the ways in which similar misdiagnoses can be avoided for clinicians in the future. Acute appendicitis is a highly prevalent condition that serves as one of the most common causes of acute abdominal pain with a lifetime risk of 8.6% in males and 6.7% in females . It mostA 21-year-old female patient with no significant past medical history presented to the Emergency Department with acute onset abdominal pain for the past three days. She reported having multiple sexual partners and sexual intercourse with a new partner three days ago and immediately afterward developed crampy diffuse abdominal pain. The patient also reported the onset of non-bloody watery diarrhea associated with bowel incontinence the day before the presentation. She denied any sick contacts, recent travel, fever, chills, nausea, vomiting, and dysuria. She denied any vaginal discharge, malodor, and previous history of sexually transmitted infection (STI).9/L. Computed tomography (CT) scan of the abdomen/pelvis was obtained which showed tubular complex fluid collections in bilateral adnexal regions, suggestive of bilateral TOA  and therefore TOA, which was corroborated by CT findings . In addiImaging is a highly useful adjunct during the stage of diagnostic reasoning, however, it may not always provide the correct diagnosis and should not be treated as a substitute for clinical reasoning. In a case report presented by Taylor et al., a 47-year-old female developed a TOA that was misdiagnosed as an appendiceal abscess based on CT findings, leading to a delay in care . SimilarStudies have shown mixed results in regard to operative versus non-operative treatment of complicated appendicitis. However, a systematic review and meta-analysis of 17 studies by Simillis et al. demonstrated that non-operative management of complicated appendicitis led to a lower reoperation rate and decrease in complications while maintaining a similar length of hospital stay as compared to immediate surgical intervention . ConservAcute appendicitis in the absence of classical features can be a challenging diagnosis that mimics other conditions. Clinicians should be aware of these unusual presentations and assess the patient's overall clinical picture rather than solely depending on imaging findings, in order to avoid misdiagnoses and delays in care. Once the diagnosis of complicated appendicitis has been made, conservative treatment with percutaneous drainage and antibiotics without operative management can be initiated.


Intramuscular haemangiomas are rare, benign vascular tumours that represent < 1% of all haemangiomas. When involving the extraocular muscles, haemangiomas are extremely rare, with only nine cases reported in the literature; to date there are no reported cases of extraocular muscle haemangiomas extending into the brain.A 6-year-old boy with a two-week history of a painless swelling and erythema on the upper eyelid. On examination, the patient had restricted extraocular motility in all directions of gaze. In addition, the eye appeared proptotic, with chemosis and hyperaemia of the conjunctiva. Visual acuity and intraocular pressure were normal. Orbital computed tomography imaging showed a mass in the left superior rectus, with heterogeneous enhancement following contrast administration. Incisional biopsy revealed an intramuscular haemangioma of the superior rectus muscle with capillary-type vessels. The patient received an intralesional steroid, which improved the condition for a few months; however, the lesion later recurred and included an intracranial extension.This case represents the first reported case of intracranial extension of intramuscular haemangioma of extraocular muscle. Intramuscular haemangiomas (IMHs) are a rare type of tumour, accounting for less than 1% of the total number of haemangiomas; their occurrence in extraocular muscle is extremely rare . IMHs arA 6-year-old boy, not known to have any medical illness, presented to our hospital complaining of painless redness and swelling of the left upper eyelid with proptosis that had developed over two weeks. Upon examination, best corrected visual acuity (BCVA) of the right and left eyes were 20/20 and 20/30 respectively. Intraocular pressure (IOP) of both eyes was 12 mmHg, and the left eye showed mild upper lid swelling, proptosis with restricted extraocular (EOM) movement in all gaze directions. Examination of the anterior segment shows mild chemosis with hyperaemia, clear cornea and normal fundus  scan with contrast revealed a left orbital soft tissue mass measuring 3.7 × 1.7 cm. The isodense mass in the muscle displaced the superior rectus and left globe downwards and anteriorly. The mass showed heterogeneous enhancement. There was bone erosion to the superior left orbital bone ridge (roof) extending to the brain Fig. .Fig. 2OrUnder general anaesthesia, a biopsy was performed through the infrabrow region using a subperiosteal approach to the midsuperior orbit. The histopathologic analysis revealed large, variably sized vessels infiltrating between muscular fibres Fig. .Fig. 3HiA diagnosis of a capillary-type intramuscular haemangioma of the superior rectus muscle was made. The patient received one dose of intralesional triamcinolone acetate (TA) (40 mg /ml). The lesion regressed, proptosis resolved, and full extraocular movement was restored, with normal BCVA and IOP.Few months later, the patient came back complaining of the same symptoms in the same eye. Upon examination of the left eye the BCVA was 20/40, the IOP was 14 mmHg, and there was complete restriction of EOM. Another dose of TA was given, but there was no improvement. A CT scan of the orbit showed the tumour had recurred; it was accompanied by bone destruction and an intracranial extension that involved the sphenoid sinus. Furthermore, following the administration of contrast, CT revealed strong enhancement of the tumour (Fig. IMHs constitute less than 1% of all haemangiomas and are usually located in the skeletal muscles of the trunk or limbs; they rarely occur in the head and neck region . IsolateOnly nine such cases of IMHs involving extraocular muscles are reported in the literature Table . The ranChristensen et al.  were theTrichinella spiralis infection, cysticercosis and dermoid cysts. Hence, to establish a definitive diagnosis of IMHs is difficult; it is only possible with biopsy and histopathology. Imaging features of IMHs often show a sharp demarcation, with bright areas within the tumours that indicate a combination of large vessels with stagnant blood and nonvascular tissue. Images also show areas of septated-striated high signal intensity and curvilinear areas of low signal intensity [The differential diagnosis of enlarged EOM includes thyroid eye disease, idiopathic inflammation, metastases and lymphomas. Other but less likely possibilities include amyloidosis, ntensity , 6, 7.Many treatment modalities have been advocated for cases of cutaneous and extra-orbital skeletal haemangiomas; these include administering cryotherapy, radiotherapy, steroid or sclerosing agents and embolisation. However, at present, the optimal management approach remains total surgical excision that includes an adequate margin of surrounding healthy tissue , 5–7. ThThe recurrence rates of IMH after incomplete excision are reported to be 20% for the capillary type, 9% for the cavernous type and 28% for the mixed type of IMH .In our case, the superior rectus was involved; histopathological examination revealed the tumour to be of a capillary-type. Due to the unpredictable surgical results and the potential consequence of causing irreversible motility disturbance, total excision was not considered as treatment option at the time of presentation. Furthermore, our case demonstrated a recurrence of the tumour, and exceptionally, an intracranial extension, that has never been reported previously in the literature.In conclusion, IMH of the extraocular muscles are extremely rare, but it should be considered in the differential diagnosis of an extraocular muscle enlargement. The diagnosis is usually difficult to establish without biopsy. Due to its rarity, the most appropriate treatment for IMHs of the extraocular muscles remains challenging. No extension to surrounding tissues or intracranial extension was acknowledged until now.


The Klippel-Trénaunay syndrome (KTS) is a rare form of a birth disorder that includes capillary malformation, hypertrophy of bones and soft tissues, and tortuous varicosities, as well as hypertrophy of the capillaries resulting in hemangiomas and port wine discoloration. KTS is also known as angio-osteohypertrophy syndrome and dysplastic angiopathy. In this case report, we describe the case of a 13-year-old female with multiple superficial varicosities on the medial aspect of her left leg since birth. Computed tomography angiogram assessed and identified abnormal venous drainage in the lower limb. Klippel-Trénaunay-Weber syndrome (KTWS) differs from KTS in that KTWS involves arteriovenous malformations. VG5Q gene causes KTS, which results in abnormal development of the mesoderm during the intrauterine life of the fetus.Maurice Klippel and Paul Trénaunay described Klippel-Trénaunay syndrome (KTS) as a rare variant of congenital birth malformations in 1900 ,3. HemanMultiple extremities may be affected by the malformation of the capillary and superficial venous system . MalformA 13-year-old female born of a non-consanguinous marriage presented to the pediatrics department with a history of multiple dilated veins and overlying cutaneous pigmentation in the left lower limb since birth and abdominal pain on the left side  of the lower limb was advised; however, due to the family and the child being averse to undergoing an MRI, a computed tomography (CT) scan of the lower limb with an angiogram was conducted. CT of the abdomen showed an enlarged spleen measuring 13 × 5.9 cm with multiple geographical hypodense lesions seen in the splenic parenchyma along with progressive enhancements due to splenic hemangiomas Figure .CT of the lower limb with angiogram showed multiple nodular soft-tissue density lesions predominantly involving the posterior, medial intramuscular compartment of the thigh, calf muscles, and medial subcutaneous aspect of the left lower limb. Several similar lesions were also seen on the left foot. Multiple superficial varicosities were seen in the left leg. Soft-tissue calcific foci were noted around the knee joint and the calf muscles representing phleboliths Figure .On the angiogram, several of these lesions were dilated, tortuous enhanced vascular channels. There were no demonstrable arteriovenous channels. The arteries of the left lower limb showed no significant abnormality Figure .The underlying bones of the left lower limb appeared mildly osteopenic. The calf muscles appeared atrophied with fatty replacement. There was no evidence of disproportionate skeletal hypertrophy Figure .Two French doctors, Maurice Klippel and Paul Trénaunay, demonstrated that three children suffered from cutaneous hemangiomatous lesions along with asymmetrical hypertrophy of soft tissues and bony components in the early 19th century, coining the term “nevus variquece osteohypertrophique” . KTS is VG5Q and a de novo supernumerary ring chromosome 18 [There are several genetic defects that contribute to KTS, including overexpression of the angiogenic factor osome 18 . Most caosome 18 . It has osome 18 ,5.The common features encountered in KTS include discoloration of the dermal aspect of capillaries with varicosities in the Servelle vein which is a marginal vein seen subcutaneously . AccordiComplications related to vascular malformations include dermatitis due to the pooling of venous blood, thrombophlebitis, cellulitis, and worse scenarios, including occlusion of the venous system resulting in consumptive coagulopathies and saddle thrombus in the pulmonary circulation and strain over the heart resulting in its failure .Antenatal ultrasound may diagnose KTS during the late first trimester based on lower limb discrepancies with associated cystic lesions in the subcutaneous plane, fetal hydrops, and polyhydramnios ,10,11. AKTS is characterized by capillary and venous malformations and limb overgrowth, as well as lymphatic malformations. A thorough imaging evaluation is essential to recognize the unusual distribution of varicosities and assess the full extent of the disease. Identifying and treating further complications is part of the further management of the disease.


Congenital insensitivity to pain with anhidrosis (CIPA) is a rare, autosomal recessive disease classified as hereditary sensory and autonomic neuropathy type VI. Patients with CIPA are characterized by insensitivity to pain, episodes of unexplained fever, anhidrosis, self-mutilating behavior, intellectual disability, and autonomic nervous system abnormalities. The clinical features may intrinsically pose anesthetic challenges. We present a case of a patient with CIPA who underwent tumor biopsy under general anesthesia using a Supreme laryngeal mask airway without any complications. The anesthetic management of this condition is discussed. Congenital insensitivity to pain with anhidrosis (CIPA), also known as hereditary sensory and autonomic neuropathy type IV (HSAN IV), is a rare, autosomal recessive disorder . The cliNTRK1) gene, and CIPA was diagnosed when he was 7 years old. Other characteristics of the patient were intellectual disability, dry skin, unsteady walking, swelling of the left hip, and short left lower extremity. His preoperative hemoglobin was 8.8 g/dl. Ultrasound and magnetic resonance imaging demonstrated dislocation of the left hip joint and a cystic and solid mass around the left hip joint, approximately 10.5 cm × 5.9 cm in size, with abundant blood flow inside, and Ewing's sarcoma with aneurysmal bone cyst was considered. A diagnostic tumor biopsy under general anesthesia was scheduled.An 8-year-old boy, height 135 cm, weight 24.5 kg, was admitted to our hospital with a fever lasting more than 12 days. His body temperature ranged from 38.5 to 40 °C, with fever peaks twice daily. He was discovered to be insensitive to pain and unable to sweat after birth. Genetic testing revealed a mutation of the neurotrophic tyrosine receptor kinase 1 . His tympanic temperature was 37.0 °C. The restless patient was sedated with intravenous midazolam 1 mg and dexmedetomidine 1 µg/kg infused within 10 min. Anesthesia was induced with intravenous propofol 50 mg, sufentanil 2 µg, and sevoflurane 2% inhaled NTRK1 gene, which encodes tropomyosin-related kinase receptor A, a high-affinity receptor for the nerve growth factor (NGF) (NTRK1 gene cause apoptosis of NGF-dependent nociceptive sensory neurons and autonomic sympathetic neurons during embryonic development, leading to a lack of nociceptive reception (Congenital insensitivity to pain with anhidrosis is an autosomal recessive disease caused by mutations in the eception . PatientFor patients who can cooperate, neuraxial anesthesia can be used for abdominal and lower extremity surgeries. Pirani et al.  reportedDespite a lack of peripheral pain sensation, CIPA patients still respond to airway manipulation, and a certain depth of anesthesia is needed to reduce the hemodynamic response to endotracheal intubation . TherefoDue to autonomic nervous system dysfunction, CIPA patients may be predisposed to gastroparesis, delayed gastric emptying, and an increased risk for regurgitation and aspiration. Zlotnik et al.  reportedA primary anesthesia concern with CIPA is impaired body temperature control. Although hyperthermia is seldom reported in the literature, body temperature should be carefully monitored and controlled with cooling or warming blankets if necessary . The ope2-agonist with sedative and analgesic effects and minimal respiratory depression. These favorable characteristics make it potentially useful for anesthesia in pediatric surgeries (Dexmedetomidine is a highly selective αurgeries , 11. In This case demonstrated that for patients with congenital insensitivity to pain with anhidrosis, the Supreme LMA and dexmedetomidine-sevoflurane-based anesthesia might be safely used for minor surgery. Body temperature and end-tidal carbon dioxide should be routinely monitored.


Although the relationship between androgens and hepatocellular tumor development has been noted since 1975, cases involving hepatocellular carcinoma (HCC) or cholangiocarcinoma development in patients on chronic androgen therapy or anabolic androgenic steroid (AAS) use are few, and far between. We present three cases of patients who developed hepatic and bile duct malignancies in the setting of AAS use and testosterone supplementation, arising from a single tertiary referral center. Additionally, we review the literature for the mechanisms behind the possible androgen-mediated malignant transformation of these liver and bile duct tumors. The relationship between androgens and hepatocellular tumor development was first noted in 1965 in patients with aplastic anemia who were treated with androgen therapy ,3. In thThis article was previously presented as a meeting abstract at the 2021 ACG Annual Scientific Meeting on October 22-27, 2021 in Las Vegas, NV.Case 1A 32-year-old male, a professional bodybuilder, presented to the emergency room with severe right upper quadrant pain. He had been using AAS as well as D-BOL (methadrostenol), a nutritional testosterone supplement marketed toward bodybuilders for four years and had stopped taking both one month prior when routine blood tests revealed elevated liver enzymes. He had no known liver disease or risk factors for it. Physical examination was unrevealing with no notable hepatomegaly or stigmata of chronic liver disease. Laboratory evaluation revealed alanine transaminase (ALT) 46 IU/L, aspartate transaminase (AST) 101 IU/L, alkaline phosphatase 41 IU/L, and total bilirubin 1.2 μmol/L. Tumor markers were within normal limits  level of 1.3 ng/mL and a carcinoembryonic antigen (CEA) level of 0.9 ng/mL). Hepatitis C virus, Hepatitis B virus, hemochromatosis, autoimmune hepatitis, and all other possible causes of liver disease were ruled out. An abdominal sonogram showed no evidence of hepatic steatosis. Computed tomography (CT) scan of the abdomen showed a large, complex, indeterminate mass in the left hepatic lobe and subtle nodular foci in the right hepatic lobe. Magnetic resonance imaging (MRI) of the abdomen revealed 5 dominant, LI-RADS 5 lesions with arterial enhancement and washout as well as numerous arterially enhancing lesions with variable washout throughout the liver  for over 20 years presented to the emergency room with right upper quadrant pain and found to have a large liver lesion in the right hepatic lobe on CT scan of the abdomen. Laboratory evaluation revealed ALT 42 IU/L, AST 58 IU/L, alkaline phosphatase 69 IU/L, and total bilirubin 1.5 μmol/L. AFP was significantly elevated at 4,326 ng/mL and peaked at >18,000 ng/mL. MRI of the abdomen showed a dominant segment 8 LI-RADS 5 lobulated hepatic mass with arterial enhancement and washout and adjacent satellite nodules as well as additional segment 7 and segment 6 arterially hyperenhancing lesions with variable washout and extensive tumor thrombus in the portal venous system  was markedly elevated at 13,000 U/mL. A CT of the abdomen demonstrated a large central hepatic mass measuring 8.4 x 8.4 x 3.2 cm with too numerous to count masses scattered throughout the liver  . Other sClinicians should have a low threshold to biopsy hepatic adenomas with suspicious imaging findings such as peripheral rim enhancement or contrast washout. Furthermore, on the basis of this strong correlation between AAS therapy and the development of HCC, we propose routine screening for tumor progression in at-risk patients who have been found to have a hepatic adenoma with US imaging and AFP level every six months.Cases such as these emphasize the need to raise awareness of potentially fatal adverse effects in the medical and bodybuilding communities as well as in patients suffering from hypogonadism, aging men, and patients on long-term estrogen and other steroid therapy. It is critical for clinicians to be vigilant in monitoring patients receiving testosterone supplementation or other androgen and estrogen therapies for potentially life-threatening complications such as HCC and cholangiocarcinoma. Heightened regulation and public health measures are needed in bodybuilding communities to provide education regarding the risk of carcinoma development linked to the use of AAS.Additional research is needed to better understand the pathogenesis of tumor development in patients on AAS or testosterone-replacement therapy. Future research studies such as prospective cohort studies to observe outcomes of liver and bile duct cancers in subjects taking steroid supplementation may be warranted to better define the relationship between the use of these substances and tumor development. Future directions should focus on the development of universal guidelines to screen for hepatocellular and bile duct cancers in patients on long-term AAS therapy.


We report a 48‐year‐old female patient with various stress fractures of extremities, musculoskeletal pain, and tooth loss. Hypophosphatasia was diagnosed based on clinical and laboratory findings and ALPL genetic results. This case highlights the importance of early diagnosis of hypophosphatasia in adults and appropriate treatment to prevent further complications. Adult hypophosphatasia may present with recurrent fractures, chronic musculoskeletal pain, tooth loss, and low alkaline phosphatase level. Asfotase alfa therapy can be used to improve respiratory function, motor function, and reduce tooth loss. Past medical/surgical history also included perimenopause, nasal cartilage collapse, breast reduction, and 4 bone grafts. She was from European descent, and family history was notable for a mother with early adulthood hip fracture and osteoporosis. She denied any use of tobacco, alcohol, and did not exercise regularly due to pain and limited range of motion. On physical examination, vital signs, heart, and lungs were normal. Dental examination demonstrated loosen teeth of both upper and lower jaws confirmed on X‐rays Figure  and diff2Answer: Hypophosphatasia.Dental findings and medical history are all consistent with the diagnosis of juvenile onset hypophosphatasia. Hypophosphatasia is characterized by defective mineralization of bone and/or teeth and low serum and bone alkaline phosphatase.Lindsay Thi Hoang: Conceptualization; data curation; formal analysis; investigation; writing – original draft. Jennifer S Hatfield: Conceptualization; formal analysis; investigation; writing – review and editing. Quan Nguyen: Conceptualization; investigation; writing – review and editing. Mohamed KM Shakir: Conceptualization; formal analysis; investigation; resources; supervision; writing – review and editing. Thanh D Hoang: Conceptualization; formal analysis; investigation; methodology; resources; supervision; validation; writing – review and editing.None to declare.The manuscript has been reviewed and approved by the IRB and Public Affairs Office.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


An 18-year-old boy presented with a giant midline mass with 9 years of evolution. The tumor was excised, and reconstruction made with a customized sternum implant and a free latissimus dorsi muscle flap with skin graft. Histological analysis was compatible with low-grade fibromyxoid sarcoma (LGFMS). Low grade fibromyxoid sarcoma (LGFMS) was first described by Evans in 1987 [LGFMS is an extremely rare malignant tumor that appears in young adults, with a reported incidence of 0.18 per million ,3. It coLGFMS usually presents as a painless, slow-growing mass, within the deep tissues of the extremities and trunk, but rarely arising primarily from the chest wall. It has been described in other unusual locations such as the head and neck or abdominal cavity. Metastasis can be present, usually late in the disease, mainly to the lung .Herein we report a case of a giant thoracic LGFMS with origin on the sternum.An 18-year-old boy was evacuated from his home country (Guinea-Bissau) to Portugal because of a giant midline thoracic mass, that had been slowly growing for the last 9 years . The patA CT scan was made which identified a 17 × 17 × 12 cm heterogeneous mass with signs of disperse calcification, arising from the body of the sternum . SeveralA combined Thoracic Surgery and Plastic Surgery approach was planned, and removal of the complete sternum was agreed upon. Therefore, a custom-made thoracic implant was designed based on the CT scan.For the surgery, patient was placed supine and the mass was excised respecting 2 cm of macroscopic margins, with intra-operative finding of pericardial invasion by the tumor. Final defect was 20 × 20 cm. After surgical excision, a pericardial mesh was used to reconstruct the pericardium and the sternum prosthesis fixed to the remaining ribs  and 4. IHistological and immunohistochemical analysis revealed LGFMS with free margins, and exclusive and multifocal MUC4 expression, respectively.At 6 months of follow-up the patient presented no signs of recurrence and has no physical activity restraints .The majority of chest wall tumors are malignant and arise from metastasis or direct invasion from adjacent tumors of the thorax, mediastinum or soft tissue, more frequently from breast and lung cancers. Primary chest wall tumors are relatively uncommon . Around LGFMS is a malignant tumor which often misleads pathologists for its innocuous histological appearance. It was first described by Evans and has been since then accepted as an individual entity.The tumor is more common in young adults (median age of 32.5 years), even though it can affect all ages and almost one fifth of the cases may present in the pediatric age ,9.Our case appears to have origin in sternum-chondral junction with posterior growth into the thoracic cavity. Other reports of chest wall LGFMS have been reported with a variable clinical spectrum – from asymptomatic growing tumor to persistent cough or acute respiratory distress ,5,9–14. Pediatric patients usually present with more superficial forms of LGFMS and appear to have better prognosis. Small size (<3.5 cm) also seems to a good prognosis factor .FUS and CREB3L2 genes, with its resulting chimeric protein present in 95% of well-defined LGFMS [Histologically, LGFMS is characterized by bland appearing spindle cells in a patternless or whorled growth pattern in a combination of myxoid to highly collagenous/fibrous stroma. Mitotic figures are absent or rare, and cellularity is typically low even though myxoid areas present more cellularity compared to the fibrous ones. The former also present a more developed vascular network . Pericoled LGFMS ,9,12–14.Treatment consists of surgical resection with clear margins. Chemotherapy and radiotherapy have been used, particularly in positive resection margins, but its efficacy is still up to debate ,3. One oStarPore™ – Anatomics™), which is significantly lighter and more flexible. It also allows intraoperative modification and fast tissue integration [StarPore™ prostheses, manufactured from a preoperative CT of the patient. For soft tissue reconstruction a latissimus dorsi muscle free flap was chosen as it presented adequate size and thickness to fully cover the prosthesis and the soft tissue defect.Sternal reconstruction is essential for protection of the mediastinal contents, stabilization of the thorax and for maintenance of respiratory physiology. Usually, titanium plates or meshes are the chosen method for reconstruction but molding of the plates is dependent on surgeon’s experience. Recently, there has been an interest in developing 3 D printed custom-made prostheses for total sternum reconstruction . These pegration . We haveRecurrences and metastases have been described in the literature, usually ranging from 9 to 21% and 6 to 27%, respectively ,17. HoweWe present a rare case of LGFMS in an unusual location. These tumors should be considered in the differential diagnosis of deep-seated masses of the thoracic wall, especially if long-term growth is noted. Correct diagnosis is of particular importance as long follow-up must be considered is these patients.


Imaging may be helpful for determining the correct diagnosis. Surgery represents a potentially curative treatment of RLPS.Liposarcoma (LPS) represents the most common type of retroperitoneal sarcoma (RPS) and can be classified into four subtypes. Preoperative diagnosis of retroperitoneal liposarcoma (RLPS) is a challenge because of its A 55-year-old Caucasian female presented to the Emergency Department with a two-day history of abdominal pain, abdominal distension, inability to pass gas or stool, nausea, vomiting and lipothymia. Abdominal examination revealed abdominal distention, abdominal pain without obvious muscle guarding and a giant non-tender mass. Laboratory tests reported neutrophilic leukocytosis and anemia. Abdominal contrast-enhanced computed tomography (CECT) showed a heterogeneous and hypodense giant retroperitoneal mass compressing and displacing the surrounding organs and vessels. The patient underwent excision of a giant retroperitoneal mass. The postoperative course of the patient was uneventful.RLPS is a malignant neoplasm that can slowly grow to enormous size with possible involvement of adjacent organs and vessels; it may recur locally and has a minimal capacity to metastasize. Preoperative diagnosis and staging of RLPS are important to establish appropriate management and prognosis. Surgery represents the gold standard for non-metastatic RLPS treatment.late and nonspecific clinical presentation. CECT represents the most commonly used modality for diagnosis, staging and preoperative evaluation. Surgery represents the appropriate treatment of non-metastatic RLPS.RLPS is a rare malignant neoplasm generally difficult to detect early due to its  •Liposarcoma is the most common type of retroperitoneal sarcoma.•clinical presentation.Early diagnosis of retroperitoneal liposarcoma is a challenge due to its nonspecific •Computed tomography and magnetic resonance imaging are the most relevant imaging modality for diagnosis•Surgery represents a potentially curative treatment of retroperitoneal liposarcoma. Preoperative diagnosis and staging are important to establish appropriate management and prognosis. A rare case of giant RLPS, presenting in emergency with intestinal occlusion, is presented with review of the literature in accordance with SCARE 2020 criteria Retroperitoneal sarcomas (RPSs) are rare malignant neoplasms that account for approximately 12–15% of all soft tissue sarcomas, with a mean incidence of 3–4/million 23/μL) and anemia (hemoglobin 7.5 g/dl). The patient was initially managed with fluids, transfusion of three units of packed red blood cells, intravenous broad-spectrum antibiotics and bowel rest. Abdominal contrast-enhanced computed tomography (CECT) showed a heterogeneous and hypodense giant retroperitoneal mass, measuring 36 × 32 × 28 cm, compressing and displacing the surrounding intra- and retro-peritoneal organs and vessels and suspected of retroperitoneal soft tissue sarcoma   . The pat3RPSs are rare neoplasms  4Retroperitoneal WDLPS is a rare malignant tumor characterized by slow progressive growth, local invasion and local recurrence after surgery but does not exhibit metastatic potential. Preoperative diagnosis and staging is essential for optimal management of RLPS. Surgery represents the mainstay of treatment for non-metastatic RLPS.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Ethical approval has been exempted by our institution because this is a case report and no new studies or new techniques were carried out.Written informed consent was obtained from the patient, for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Giuseppe Evola: Drafting the manuscript, literature research.Riccardo Schillaci: Operated on the patient, drafting the manuscript.Martina Reina: Drafting the manuscript and literature research.Giovambattista Caruso: Operated on the patient, drafting the manuscript.Maria D'Angelo: Drafting the manuscript, literature research.Giuseppe Angelo Reina: Operated on the patient, revising the manuscript.Not applicable.Giuseppe Evola.Not commissioned, externally peer-reviewed.All the authors certify that there is no conflict of interest regarding the material discussed in the manuscript.


Meningioma is the most common intracranial neoplasia in cats. Treatments for meningiomas—including complete surgical resection, debulking, irradiation, or palliative therapy—have been reported in veterinary medicine. However, multiple meningiomas  have been reported to affect the complication rate and prognosis. Moreover, the characteristics of neurosurgery—such as accurate localization and awareness of the anatomical structures of the lesions—make the surgery especially difficult for inexperienced surgeons. Surgical navigation systems have been developed, but recently, patient-specific three-dimensional(3D)-printed models and guides have also been used in orthopedics and neurosurgeries for treating many disorders with good results. A 13-year-old castrated male domestic shorthair cat was referred with multiple meningiomas located within the right frontal and occipital lobes. The cat suffered from generalized tonic–clonic seizures and mild proprioceptive ataxia. After removing both of the tumors, the cat showed a favorable clinical outcome and no neurological abnormalities throughout long-term follow-up. With a patient-specific 3D guide technology, a craniotomy for multiple meningiomas can be performed safely and accurately.A 13-year-old castrated male domestic shorthair cat was referred for the surgical removal of multiple meningiomas. The cat experienced generalized tonic–clonic seizures, altered mentation, mild proprioceptive ataxia, and circling. Magnetic resonance imaging (MRI) revealed two round, solitary, well-delineated, space-occupying lesions suggestive of multiple meningiomas in the right frontal and occipital lobes. Before surgery, patient-specific three-dimensional (3D) printed models and guides were produced using a 3D program based on MRI and computed tomography (CT), and a rehearsal surgery was performed. With a 3D guide to find the location of the craniotomy lines, bilateral extended rostrotentorial craniotomy allowed en bloc resection of multiple meningiomas. The bone fragment was replaced and secured to the skull with a craniofacial plate and screws with an artificial dura. All of the surgical steps were performed without complications. The preoperative presenting signs were resolved by the time of follow-up examinations 2 weeks after surgery. Twelve months after the removal of the multiple meningiomas, the cat survived without further neurological progression. For the resection of multiple meningiomas, surgery can result in large bone defects and risk of massive hemorrhage. For this challenging surgery, patient-specific 3D models and guides can be effective for accurate and safe craniotomies. The most common intracranial neoplasia in cats is meningioma . In a reMost intracranial meningiomas are extra-axial intracranial tumors that originate from arachnoid cap cells, which are cells of the arachnoid villi in contact with the venous endothelium of the venous sinus of the dura ,4,5. TheTreatment options for meningiomas include complete surgical resection, debulking, irradiation, or palliative therapy. In previous reports, each treatment option showed significant differences in the median survival time. The median survival time for cats following various treatments were as follows: surgical resection, 26 months; irradiation therapy, 14 months; and medical management, 18 days . Unlike Various techniques have been developed, including surgical navigation systems (SNSs) and 3D printing models and guides. In human medicine, SNSs have been proven beneficial in intracranial surgery , and theRecently, the use of 3D-printed models and guides has also increased in human and veterinary medicine to overcome these limitations. The use of 3D-printed surgery models help surgical teams carry out rehearsal surgery, patient-specific surgical planning, and identification of the exact intraoperative location of the lesion ,15. It aA 13-year-old castrated male domestic shorthair cat was referred for surgical removal of presumed multiple meningiomas within the right frontal and occipital lobe detected during magnetic resonance imaging (MRI) at the referring veterinary clinic. Reported clinical signs included generalized tonic–clonic seizures, altered mentation, and circling. In addition, the patient was not able to jump or climb to higher places. The cat was diagnosed with multiple meningiomas 2 months before referral and was initially treated with prednisolone (0.5 mg/kg PO q12h) and hydroxyurea (25 mg/kg SID) for two months.At presentation, the patient exhibited circling and ataxia. MRI showed a round, solitary, well-delineated, space-occupying lesion with marked hypointense to isointense T1-weighted (T1W) images, hyperintense T2-weighted (T2W) images, and strong contrast-enhanced T1W post-contrast images in the right frontal and occipital lobe . It alsoSurgical planning involving confirmation of the location, size, and shape of the craniotomy line and identifying the exact location of the tumor was performed under computer-aided 3D design software  based on MRI and CT images. In addition, 3D models were produced and obtained according to the program and a previously described protocol . A 3D cr2O2 gas sterilizer ).After producing the patient-specific 3D models and guides, the rehearsal surgery was performed to familiarize the surgeon with the surgical procedure and patient-specific anatomo-pathological features. In addition, the ideal position of the patient for the procedure—i.e., sternal recumbency with the head elevated to approximately 30° without lateral tilting to promote brain “dropping” with the aid of gravity—was determined. After the rehearsal surgery, the 3D models and guide were sterilized with plasma sterilizer ), Medetomidine ), Dexamethasone ), and Alfaxalone ) were administered [®, Hana Pharm, Seoul, Korea)) was used for analgesia. Anesthesia was maintained with isoflurane ). Plasmalyte ) was administered throughout the procedure. Mannitol ) was also administered to relieve increased intracranial pressure (ICP) and to induce brain relaxation [For anesthetic premedication and induction, Midazolam ; After the craniotomy, the masses were gently mobilized by grabbing their surface with atraumatic forceps and packing lint-free gauze between the tumor and brain tissues to divide the tumor from the brain parenchyma. En bloc resection was performed with the open-window technique by enucleating the tumor with tumor forceps or surgical aspirator ; After the en bloc resection of the meningiomas, an oozing hemorrhage was observed. The hemostatic matrix was applied twice for each surgical site to control oozing hemorrhage from the surgical site. The application time for each process was 2 min, following which all matrixes were flushed out with physiological saline, and the hemorrhage was evaluated again . When he®; Samjin Pharm. Co., Ltd., Seoul, Korea)). The cat also received dexamethasone ) for 3 days to prevent surgical site swelling after surgery. Phenobarbital ), and Levetiracetam ) were also administered after surgery.Postoperatively, the cat was sedated for the first 24 h with medetomidine  . The catOn the day after surgery, the cat showed no abnormalities on physical examination. Neurological examination revealed mild proprioceptive ataxia with mildly dragging hind limbs. Menace response and pupillary light reflex were normalized just a day after surgery. The preoperative presenting signs were resolved by the time of the 2-week follow-up examination. The cat was discharged from the hospital with a normal gait and without any neurological abnormalities. Telephonic follow-ups were conducted to obtain information regarding patient status and gait evaluation. Clinical follow-up could not be performed because of the difficulties faced by the owner in visiting the hospital with the cat.At 370 days after the operation, the cat showed alert mentation, without further complications such as neurological progression or ataxia. In addition, the cat could jump and climb higher than it could before surgery. Further postoperative MRI and CT were denied by the owner due to the risk of repeated anesthesia for the patient and costs.Postoperative histological diagnosis was meningioma for both the masses. The two types of samples exhibited the following similar characteristics: moderate to dense cellularity and spindle cells arranged in bundles and a few whorls. A small amount of collagen was observed. The cells had a moderate amount of cytoplasm and elongated nuclei. The mitotic count was 1 mitoses/10 HPF. The tumors were classified as grade 1 (benign meningioma) fibroblastic subtype based on the human World Health Organization schemes, which is one of the most common subtypes of feline meningiomas .To the best of our knowledge, this is the first report to describe the resection of multiple meningiomas by bilateral extended rostrotentorial craniotomy using a patient-specific 3D guide. The 3D-printed guide perfectly fit the skull as designed, and the craniotomy was completed as planned. The accurate surgical approach makes the surgical procedure safer. The patient had excellent outcome for a long-term follow-up of 370 days without complications. The cat showed improvement in ataxia and reaction to external stimuli without seizure or ataxia after complete en bloc resection of meningiomas using the open-window technique.In human medicine, if the total resection of meningioma presents risks of further complications because of the location or adjacent anatomic structures of meningioma, subtotal resection or debulking is inevitably performed rather than complete resection ,4. HowevVarious surgical approaches and craniotomy sites are chosen based on the anatomical location of the meningioma and patient-specific considerations ,4,21. FuIn addition to an excellent craniotomy plan that determines the craniotomy localization, size, and shape, accurate craniotomy and determining anatomo-pathological relationships are crucial in neurosurgical procedures to reduce mortality and morbidity ,13. An aAlthough bilateral extended craniotomy has advantages in reducing excessive manipulation and traction of brain parenchyma, it still carries the risk of damaging the sagittal sinus and collateral and bridging veins. In this case, according to the diagnostic images, the dorsal sagittal sinus was deviated ventro-laterally as a result of the large volume of the meningioma. Furthermore, there was no significant sign of infiltration of meningioma into the dorsal sagittal sinus. These were believed to be positive factors for enhancing the surgical outcome. However, it is still challenging for the surgeon to perform bilateral extended craniotomy while preserving the dorsal sagittal sinus. To reduce the risk of dural tear and iatrogenic injury to the dorsal sagittal sinus and improve surgical outcome, brain relaxation was performed perioperatively . Brain rIn this case, despite the effort to preserve dura mater, a dural tear occurred during the craniotomy. In human medicine, the incidence of dural tears during craniotomy ranges from 20% to 30%, and may include single or both layers of the dura ,35. ThesTraditionally, in human medicine in the Simpson’s grading system of surgery of meningioma resection, the recurrence rate correlates with the extent of adjacent structures removed, including dural attachment, adjacent venous sinus, and nearby hyperostotic bone ,39,40. AOur case report has limitations regarding follow-up data because the owner did not want further diagnostic testing. It is essential to perform serial follow-up diagnostic imaging with neurological examinations to accurately assess surgical outcomes and signs of recurrence. If MRI and CT images could have been taken postoperatively, the relationship between the dura mater, plates, and artificial dura could have been assessed. In addition, although this case report showed the effectiveness of a patient-specific 3D-guide for feline meningioma resection, a case series or a prospective study is necessary to confirm the effectiveness and safety. Furthermore, rehearsal surgery with patient-specific 3D models also presents limitations because the models are based on preoperative images. Therefore, they cannot reflect the intraoperative brain distortion or surface bulging after dural opening.In this case, patient-specific 3D-printing technology contributed to favorable surgical outcomes in en bloc resection of multiple meningioma. Rehearsal surgery with patient-specific 3D models makes surgeons more familiar with anatomo-pathological relationships and accurate craniotomy localization. With a patient-specific 3D guide, a craniotomy can be performed safely and more accurately. Clear and adequate exposure of the lesion prevents excessive manipulation and traction of the brain parenchyma, resulting in successful en bloc resection of multiple meningiomas using the open-window technique in cats. Patient-specific 3D guides and models may also improve the surgical outcomes of other craniotomies.


A 74‐year‐old man was diagnosed with unresectable pancreatic cancer with obstructive jaundice. Chemotherapy with gemcitabine and nab‐paclitaxel was initiated after placement of a duckbill‐shaped anti‐reflux metal stent (D‐ARMS). A period of 1 month after D‐ARMS placement, the patient developed hematemesis and entered severe shock following emergency admission for further evaluation.Contrast‐enhanced computed tomography revealed a pseudoaneurysm in the gastroduodenal artery, coincident with the site of D‐ARMS placement, and bleeding from the same site was diagnosed. Angiography was performed, and the pseudoaneurysm was successfully treated by transcatheter arterial embolization using coils. The patient was subsequently discharged from hospital and experienced no further bleeding until his death due to an aggravation of the pancreatic cancer after 2 months. We report a case of pancreatic cancer with pseudoaneurysm after D‐ARMS placement. Endoscopic biliary stenting is the standard therapy for malignant biliary obstruction (MBO). A self‐expandable metal stent (SEMS) is frequently used for treating MBO due to cholangiocarcinoma or pancreatic cancer because of its long patency. However, various adverse events have been reported in association with SEMS, including cholangitis, cholecystitis, pancreatitis, liver abscess, and stent migration. Although the incidence is very rare, a potentially life‐threatening adverse event after SEMS placement is pseudoaneurysm formation. Exact diagnosis is extremely important in such cases. Here, we report a case of pancreatic cancer with pseudoaneurysm after the placement of a duckbill‐shaped anti‐reflux metal stent (D‐ARMS).A 74‐year‐old man with epigastralgia was referred to our hospital after a tumor of pancreatic head was detected on contrast‐enhanced computed tomography (CE‐CT) of the abdomen. Histopathological examination of a specimen from endoscopic ultrasound‐fine needle aspiration of the pancreatic head tumor revealed adenocarcinoma. CE‐CT performed again prior to surgical resection revealed liver metastasis and circumferential invasion of the main trunk of the superior mesenteric artery by pancreatic cancer, which was deemed unresectable. A D‐ARMS was placed to treat MBO due to pancreatic cancer Figure . Chemoth4/μl; hemoglobin, 6.5 g/dl; and platelet count, 33.4 × 104/μl. Serum albumin level was 1.9 g/dl. Blood urea nitrogen level was 24.1 mg/dl. Creatinine level was 1.11 mg/dl. Levels of lactate dehydrogenase, liver enzymes, and amylase were all within normal ranges. The patient was treated with fluid resuscitation and blood transfusion.A period of 1 month after D‐ARMS placement, the patient presented with hematemesis and shock vitals and was admitted to the emergency room. Laboratory data revealed: white blood cell count, 5950/μl; red blood cell count, 206 × 10CE‐CT revealed a gastroduodenal arterial pseudoaneurysm adjacent to the site of D‐ARMS placement, which had likely ruptured and caused the hematemesis Figure . Emergenet al. retrospectively compared the incidence of RBO after stent placement between D‐ARMS and conventional covered‐SEMS (CSEMS) in patients undergoing NAC for pancreatic cancer and reported the efficacy and safety of D‐ARMS.SEMS for MBO offers longer patency than plastic stents and has recently been reported as useful for preventing stent occlusion in patients undergoing neoadjuvant chemotherapy (NAC) for pancreatic cancer a relatively long time, 2–3 months before surgery.et al. recently reported the efficacy of CSEMS placement as a treatment for biliary hemorrhage.Complications associated with SEMS placement have been reported to include cholangitis, cholecystitis, pancreatitis, liver abscess, and stent migration. Pseudoaneurysm formation is a very rare complication of SEMS placement, but the potentially life‐threatening nature of this event makes accurate diagnosis and treatment extremely important. CE‐CT followed by angiography is effective for the diagnosis of pseudoaneurysm. Correct diagnosis of pseudoaneurysm from endoscopy, on the other hand, may prove difficult. In our case, diagnosis of the pseudoaneurysm by endoscopy was difficult. The first‐line treatment for pseudoaneurysm would common to try endoscopic hemostasis, such as medial CSEMS placement, and if hemostasis cannot be achieved, the next strategy of hemostasis by angiography may be considered. Widely used in splenic and hepatic pseudoaneurysms among abdominal pseudoaneurysms, TAE can be performed following angiography to avert rupture and subsequent fatal bleeding. Itoi The timing of pseudoaneurysm formation has been reported to vary from 5 days to within 1 year after SEMS placement.The mechanism of pseudoaneurysm formation following SEMS placement is thought to involve tumor progression and invasion of the artery, inflammation and weakening of the artery and bile duct walls due to cholangitis or chemoradiotherapy, with physical pressure from the radial force of the SEMS or metal wires at the SEMS tip causing necrosis in the adjacent artery, leading to perforation and bleeding into the bile duct with weak resistance. The diameter of the CBD may be also one of the important factors causing pseudoaneurysms. In our case, the diameter of CBD had 8 mm, but a 10‐mm‐diameter D‐ARMS was used. This discrepancy between the diameter of the CBD and the SEMS may be also thought to be one of the factors causing pseudoaneurysm. The most popular SEMSs have 8‐ or 10‐mm‐diameter and can easily be deployed in a dilated CBD. Although, either an 8‐ or 10‐mm‐diameter SEMS may be relatively large for the obstructed CBD which has a lumen collapsed like a pinhole and surrounded by tumor when the SEMS is deployed in its original diameter.In this report, we have described a case of pancreatic cancer with pseudoaneurysm after D‐ARMS placement for MBO, in which hemostasis was successfully achieved with TAE to save the patient's life. Although early diagnosis of pseudoaneurysm can be difficult, pseudoaneurysm formation needs to be kept in mind as a complication after SEMS placement in future treatment and diagnosis in the biliopancreatic region.None.All procedures were performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.


Inverted papillomas (IPs) are common benign sinonasal tumors characterized by a high risk of recurrence and potential for malignancy. The lateral nasal wall and middle meatus are the most common sites for IP to arise, but they can develop from any part of the Schneiderian membrane in the nose and paranasal sinuses. IPs are usually confined to one side of the nasal cavity; bilateral involvement of the nose and paranasal sinuses is extremely rare, with only a few cases reported in the medical literature in English-language.This report presents a case of a large IP that originated from the left ethmoid sinus and spread to both sides of the nasal cavity through the frontal sinus septum. This type of diffusion is extremely rare. The tumor was successfully treated with a combined endoscopic and external approach. The need for a better understanding of the pathogenesis of bilateral involvement is discussed, as well as the importance of surgical technique when treating this rare disease.Prior informed consent was obtained from the patient. This study was approved by the ethics committee of Shengjing Hospital, affiliated with China Medical University.A 53-year-old man presented to the hospital with a four-year history of progressive bilateral nasal obstruction and hyposmia with chronic headache. There was no history of epistaxis. He had undergone a nonendoscopic left endonasal polypectomy at another institution. The patient was otherwise healthy. Physical examination revealed bilateral intranasal polypoidal masses that bled readily on contact. The nasal septum had no perforation and deviated to the right side. Sinus computed tomography showed a hyperdense image occupying both nasal cavities and the bilateral frontal, bilateral ethmoid, bilateral maxillary, and left sphenoid sinuses A and B. Bilateral involvement of the nose and paranasal sinuses is extremely rare in patients with IP, with only a few cases reported in the English-language literature. An IP originating from one side of the ethmoid sinus can lead to the development of bilateral IPs via the nasal septum or other multicenter diffusion pathways.The preferred treatment of IPs is complete wide resection. Success is fully dependent upon complete resection of the IP and careful stripping of the underlying diseased mucosa. Successful endoscopic removal of IPs has been described by many authors, and its advantages include the avoidance of a facial incision or an external scar, a shorter hospital stay, less facial swelling, decreased postoperative pain, and reduced blood loss.An endoscopic modified Lothrop procedure (EMLP), which involves complete removal of the bilateral frontal sinus floor and superior septum, provides sufficient endoscopic access for most frontal sinus IPs and may obviate the need for an open approach.However, EMLP has several disadvantages in patients with disease extending to the lateral wall of a well-pneumatized frontal sinus or the anterior wall, as well as in cases of multifocal attachment.This report represents the first known case of an IP originating from the left ethmoid sinus and involving both sides of the nasal cavity via the frontal sinus septum. It is speculated that in a well-pneumatized frontal sinus with a larger frontal ostium, a unilateral ethmoid IP may spread to the contralateral nasal cavity. This study supports the conclusion that surgical resection is the preferred treatment for IP.The authors declare no conflicts of interest.


Actinomycetes are commensal inhabitants of the oral cavity and gastrointestinal tract that can acquire pathogenicity through invasion of injured mucosa. Appendix and ileocecal regions are the most common sites involved in abdominal actinomycosis. We report a case of unusual site actinomycosis, in the recto-sigmoid colon, presenting with abdominal pain and diarrhea after one year of partial sigmoid colectomy. A colonoscopy was done, which showed a 21 mm polypoid partially obstructing mass in the recto-sigmoid colon. Histopathology showed granulation tissue with severe acute inflammation, fibrinopurulent debris with areas of abscess, and branching filamentous organisms with sulfur granules consistent with actinomycosis. Abdominal actinomycosis can infect almost all organs within the abdominal cavity; however, it is more common around the ileocecal region. Actinomycetes are inhabitants of the oral cavity and gastrointestinal tract. Nearly 50% of cases of actinomycosis are caused by predisposing factors such as disruption of the mucosa, foreign bodies, and immunosuppression . The remaining 50% of cases are reported to be idiopathic . AppendiA 57-year-old Hispanic male with a past medical history of hypertension, diabetes mellitus, chronic kidney disease, and complicated diverticulitis status post elective partial sigmoid resection one year ago presented with a one-month history of persistent abdominal pain, nausea, and greenish diarrhea. He denied any hematochezia, melena, mucous in stool, or vomiting. On review of systems, the patient denied chest pain, shortness of breath, dizziness, fever, chills, cough, and hematuria. The patient denied any use of alcohol, smoking, or recreational illicit drugs. He denied any pertinent family history. He was afebrile 98.3°F), with a pulse of 70 beats per minute, blood pressure of 119/65 mmHg, respiratory rate of 18 breaths/minute, and O2 saturation of 97% on room air. The physical exam showed a soft non-distended abdomen and normoactive bowel sounds. However, the patient was positive for mild left lower quadrant tenderness with no rebound tenderness or rigidity. His hematological investigations were unremarkable except for creatinine level of 3.0 mg/dL, blood urea nitrogen (BUN) of 51 mg/dL (patient’s baseline), hemoglobin of 12.5 gm/dL, and mean corpuscular volume (MCV) of 91 fL. Colonoscopy showed a 21 mm polypoid partially obstructing mass in the recto-sigmoid colon distal to the patent end-to-end colo-colonic anastomosis (22 cm from the anus) . The remaining 50% of cases are reported to be idiopathic [Actinomyces remains unclear [Disruption of the gastrointestinal mucosa from diverticulitis, surgical trauma, perforation, and colitis results in the predisposition of abdominal actinomycosis . We repoiopathic . The pat unclear . Abdomin unclear . Abdomin unclear .Actinomyces creates a slowly growing inflammatory mass that results in nonspecific signs and symptoms. Similar to our patient, the initial presentation typically manifests itself with abdominal pain, nausea, and altered bowel habits. These nonspecific findings make differential diagnoses of abdominal actinomycosis more challenging, as more common diagnoses must be ruled-out  [An insidious presentation is common as the infection from disease) .Colonoscopy is useful in ruling-out malignancy or other inflammatory pathologies in these cases and confirming the diagnosis with histological testing of the collected specimens . Since aAntibiotics have shown to be effective in uncomplicated cases. Penicillin or cephalosporin is taken at high doses for at least six months or until the effective dissolution of the lesion remains the standard treatment. Clinically significant treatments have also been reported using tetracycline, erythromycin, chloramphenicol, clindamycin, and imipenem and become especially useful in patients with penicillin allergy. When combined with surgical resection, a cure rate of approximately 90% can be achieved [Abdominal actinomycosis is not a common intra-abdominal infection and requires a high level of suspicion, especially in high-risk patients such as those with a history of previous abdominal surgery. Long-term antibiotic treatment is usually an effective treatment for such cases, and surgical drainage may be required in complicated cases.


The widespread use of imaging methods has led to an increased identification of asymptomatic Pancreatic Cystic Lymphangiomas (PCL), a rare entity for which available information is very limited.We present the case of an asymptomatic 61-year-old male, submitted to elective enucleation of a pancreatic head PCL at our institution. After four years of follow-up the patient is doing well and has no clinical or imaging signs of recurrence.Though rare, PCL should be included in the differential diagnosis of pancreatic cystic neoplasms. All efforts should be made to ascertain a preoperative diagnosis, as expectant follow-up could be a reasonable approach in asymptomatic patients and/or poor surgical candidates. In the face of an uncertain diagnosis, complete surgical excision may be the treatment of choice.The medical community worldwide should be encouraged to report all cases of PCL, as to increment the overall knowledge about this lesion. •Widespread use of cross-sectional imaging increased identification of asymptomatic pancreatic cystic lesions.•Pancreatic Cystic Lymphangioma is one of those rare cystic neoplasms for which available information is very limited.•Definitive diagnosis often requires pathologic examination of surgical specimen, immunohistochemically with positivity for D2-40.•En bloc resection with negative margins is curative, however, treatment for asymptomatic cystic lymphangiomas is still controversial. ComputeEndoscopic ultrasound (EUS) further revealed absence of associated wall thickening, mural nodules, solid masses or connection with main pancreatic duct. A 22-gauge EUS-FNA was performed to aspirate approximately 5 mL of a yellow-white thick fluid, with CEA level of 0,6 ng/mL, Amylase level of 297 U/L; a triglyceride level was unable to be obtained due to partial solidification of material prior to analysis. Cytology was negative for malignancy and showed only scattered mature lymphocytes and no epithelial cells.Given the volume of the lesion, uncertainty of the diagnosis and patient's preference, he was admitted for surgical exploration, performed by hepatobiliopancreatic surgeon. After a right subcostal incision, with no evidence of locally advanced or metastatic disease, we proceeded to enucleation of the lesion. Fresh frozen sections confirmed benign disease and complete resection. Pathology report a multilocular tumour with 2 to 3 cm cysts filled with yellow-white bloody fluid and, microscopically , Fig. 4,Patient had uneventful postoperative course in the surgical ward, with mobilization and oral intake initiated in first postoperative day, discharged at the 7th postoperative day. Four years of follow-up have now been completed with no evidence of recurrence or other complaints.3Pancreatic lymphangiomas comprise <1 % of all lymphangiomas PCL is more commonly located in the body and tail of the pancreas The clinical presentation is nonspecific, sometimes related to compression of neighbouring organs, often including vague abdominal pain and a palpable mass. Pedicle torsion, rupture, infection or haemorrhage into the lymphangioma may cause acute abdominal symptoms. As in our case, a growing number of patients reported were asymptomatic with incidental diagnosis of lymphangioma on physical examination or radiographic studies for unrelated diseases The cystic nature of the lymphangioma encompasses a broad differential diagnosis .Table 1NSignificant laboratory abnormalities – including serum amylase, CA19.9 and CEA – are absent Preoperative diagnosis using EUS-FNA has been reported, based on: gross appearance of the aspirated fluid (chylous); elevated triglyceride levels (<3000 mg/dL) in the cyst aspirate, with or without the presence of numerous lymphocytes; or the characteristic tissue architecture on paraffin embedded cell-blocks from the aspirates Definitive diagnosis of PCL often requires pathologic examination of surgical specimen En bloc resection with negative microscopic margins is curative 4In this study, we report a very rare case of cystic lymphangioma of the head of the pancreas. Despite being benign and mostly asymptomatic, we believe that it should be considered in the differential diagnosis of cystic tumors of the pancreas. Complete surgical excision has the dual advantage of allowing definitive diagnosis and treatment, however, asymptomatic cases with no suspicion of malignancy in the diagnostic study, observation with clinical and radiologic follow-up may be favoured.Authors did not receive funding for this research.Not applicable.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not applicable.Miguel AlmeidaNot commissioned, externally peer-reviewed.All authors namely, Dr. Rui Quintanilha, Dr. Vitor Carneiro, Dr.ª Joana Mendes, Dr. Tiago Rama e Dr. Miguel Almeida were involved in the management of this patient. All authors contributed equally to the design of the work and acquisition, analysis and interpretation of data. Dr. Miguel Almeida and Dr. Tiago Rama drafted the manuscript. Dr. Rui Quintanilha, Dr. Vitor Carneiro and Dr.ª Joana Mendes substantively revised it. All authors read and approved the final manuscript.The authors declare that they have no competing interests.


An Inflammatory myofibroblastic tumor, a neoplasm of intermediate biological potential, of the small bowel mesentery, is a rare tumor, most commonly reported in but not confined to the pediatric age group.This case report underlines a case of a  mesentery IMT in an adult female presenting with recurrent symptoms similar to acute appendicitis.It can present with symptoms similar to acute appendicitis necessitating a high index of suspicion for its prompt diagnosis. Treatment primarily includes surgical resection with recent advances in targeted therapy with tyrosine kinase inhibitors showing promising results.IMTs can present as clinical as well as histopathological mimickers of a variety of diseases especially in the abdomen. Prompt diagnosis requires both imaging and histopathological examination. •Inflammatory myofibroblastic tumor is a neoplasm of intermediate biological potential of the small bowel mesentery.•Prompt diagnosis requires both imaging and histopathological examination.•Surgery still remains the main modality of treatment. Althoug2A 50-year old female presented to the surgical OPD with a complaint of an intermittent abdominal pain located in the right lower quadrant since last 1 year. The pain was dull and aching in nature and had increased in severity for the last 3 days. She had a history of a similar episode 6 months back when she was diagnosed to have an appendicular lump which was managed conservatively in another center. This time, she presented with a similar type of abdominal pain to a different center where she was evaluated and diagnosed to have appendicular lump and was referred to our center for further management.The patient did not have complaints of weight loss or bleeding per rectum. Her appetite was normal. No other significant history was elicited. Her vitals were within normal limits and she had no pallor, icterus, edema, cyanosis, clubbing, or dehydration. On further evaluation, an ill-defined mildly tender mobile lump was palpable in the right lower quadrant of the abdomen. Her laboratory works were unremarkable except for a hemoglobin level of 9 gm %. A Contrast Enhanced Computed Tomography (CECT) scan was hence planned for further evaluation see .The CT scan revealed a large heterogeneous mesenteric mass measuring 7.6*3.9 cm in the right lower abdomen, containing multiple enlarged lymph nodes with traversing mesenteric vessels abutting the medial aspect of the proximal ascending colon and caecum with surrounding mesenteric fat stranding densities and minimal fluid collection extending along the ascending colon up to the sub hepatic space . There wWith the above findings, the patient was planned for an elective laparotomy. Intra operatively an 8*4 cm firm, well defined lump was palpable in the right lower abdomen within the ileocolic mesentery and overlying traversing vessels were identified . The masExploratory laparotomy with extended right hemicolectomy with ileotransverse anastomosis was done. Her postoperative course was uneventful and the patient was discharged satisfactorily on the fourth postoperative day.Finding of the specimen on gross examination showed a single well circumscribed solid firm mass within the mesocolon measuring 9*7*2 cm. Cut section of the specimen revealed a solid white homogenous area with central hemorrhage. The mass was 25 cm away from the colon and 5 cm away from the ileal resection margin. Three lymph nodes were identified during the procedure. Histopathological report of the mass showed the mass was hyper cellular with cells arranged in a compact fascicular and diffuse pattern in a myxoid background. Individual cells were spindle-shaped, round and elongated with abundant eosinophilic cytoplasm. Nuclei were oval to elongated in shape without atypical features and with occasional mitotic figures. There was a dense inflammatory cell infiltration predominantly consisting of plasma cells, eosinophil, neutrophils and lymphocytes. Mature looking adipocytes were also noted . All the3Inflammatory myofibroblastic tumor is a rare mesenchymal tumor with unknown cause that encompasses a spectrum of myofibroblastic proliferation along with a varying amount of inflammatory infiltrates like lymphocyte, plasma cell and eosinophils . IMTs ocIMTs can involve almost every organ of the body. While the lungs are the most commonly involved site, IMTs have also been described in the mesentery, omentum, intestine, rectum, appendix, retroperitoneum, gastro-esophageal junction, mediastinum, liver, and abdominal wall. Among these, the most common extrapulmonary sites are the mesentery and the omentum as described in the above case presentation. Such extra-pulmonary IMTs are, however, more common among children rather than in adults . Other spolyp, and other fibro-inflammatory processes (or pseudo neoplasms) and malignant tumors [IMTs of abdomen have many differential diagnosis some of which include reactive/reparative lesions and mesenchymal tumors of the gastrointestinal tract including granulation tissues and nodular fasciitis in reactive processes, spindle cell sarcomas, peripheral nerve sheath tumors spindle cell melanomas, and sarcomatoid carcinomas, dedifferentiated liposarcoma, gastrointestinal stromal tumors, Hodgkin's lymphoma, inflammatory fibroid t tumors .Despite the many speculations into the cause of IMTs, a complete resection of the tumor remains the mainstay of its treatment. Surgery is the most accepted treatment. If there is a biopsy-proven diagnosis of IMTs that excludes malignancy, medical treatment with no steroidal anti-inflammatory drugs in patients with peripheral hepatic IMTs could be prescribed . AdjuvanThere have been recent advances in the treatment of IMTs with immunohistochemistry identifying ALK positive tumors. Such tumor have the potential of being treated with tyrosine kinase inhibitors targeting ALK which includes drugs like crizotinib and ceritinib .4IMTs can present as clinical as well as histopathological mimickers of a variety of diseases especially in the abdomen. Prompt diagnosis requires both imaging and histopathological examination. Despite recent advances in the pathophysiology of the disease, surgery still remains the main modality of treatment.Not commissioned, externally peer-reviewed.None.No funding was received for the study.SS, AS, and SKY wrote the original manuscript, reviewed, and edited the original manuscript. SKY, HBB, SS, AS, SS, AS, BKY, SJ, KS, and BBS reviewed and edited the original manuscript.Authors have no conflict of interest to declare.1.Name of the registry: None2.Unique Identifying number or registration ID: None3.Hyperlink to your specific registration (must be publicly accessible and will be checked):Sangam Shah.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.


Spondylotic myelopathy involves chronic spinal cord compression from degenerative spine changes presenting a myriad of neurological and pain symptoms. We report a case of cervical myelopathy with transverse pancake‐like gadolinium enhancement seen on MRI in a 42‐year‐old gentleman with progressive bilateral upper extremity numbness, tingling, and impaired gait. Although it is primarily a clinical diagnosis, the use of advanced imaging, such as magnetic resonance imaging (MRI), allows us to shuffle through various causes of myelopathies rapidly. Transverse pancake‐like gadolinium enhancement is a rare but specific finding on MRI which points toward spondylotic myelopathy.2A 42‐year‐old male patient with no significant past medical history presented with approximately 1 year of progressive bilateral upper extremity numbness, tingling, and impaired gait. His symptoms started when he was rear‐ended while driving his car a year prior. Approximately 2 weeks after the car accident, he began to notice numbness and tingling of his fingertips, progressing up his right upper extremity to the shoulder. He then experienced numbness and tingling of his left fingertips, which progressed proximally in a similar pattern. Subsequently, the symptoms involved his bilateral lower extremities. The lower extremity symptoms were associated with difficulty ambulating, especially lifting his left leg off the ground and occasional buckling of both knees causing him to fall. He also endorsed pain at both hips and over his neck musculature but denied pain elsewhere, including his back. With regard to sensation in his hands, only pressure sensation was spared. His only additional complaint was urge urinary incontinence. His slow gait exacerbated this problem. He had no issues with his bowel function.Physical examination was notable for generalized hypertonia (Left > Right), spasticity, hyperreflexia in all four extremities, positive Hoffman sign in both hands, myoclonus in bilateral ankles, and loss of pinprick sensation in bilateral upper extremities.Tests for human immunodeficiency virus (HIV), thyroid stimulating hormone (TSH), erythrocyte sedimentation rate (ESR), creatine kinase (CK), and syphilis were all unremarkable. Computed tomography (CT) of the cervical spine did not show any fracture or dislocation but did show left‐sided foraminal stenosis at the C3–C4 level. Cervical MRI showed a long cord section of edema and a “pancake enhancement” pattern as illustrated in Figures Hence, due to signs of cord inflammation seen on MRI and the fact that steroids have been tried even in such cases that were compressive triggered pancake sign enhancement,3Myelopathy, encompassing traumatic, vascular, neoplastic, neuropathic, infectious, and any other insult to the spinal cord remain a clinical diagnosis, with specific neurological symptoms and signs that help localize the insult to the spinal cord. Classical upper and lower motor neuron signs are prominent depending on the level of insult. Our patient, having cervical myelopathy, had upper motor neuron (UMN) signs and symptoms in both upper and lower extremities and bladder involvement. Furthermore, a “sensory level” with a band‐like sensation at the superior margin is expected in thoracic and lumbar myelopathies. However, this was absent in our patient. Hoffman's sign was positive, corroborating its frequent association with cervical myelopathy.Regarding imaging findings, pancake enhancement of the cervical spine was first reported in 2014 by Flanagan et al.4The pancake enhancement phenomena on MRI in cervical spondylotic myelopathy is very rare, with only a few other case reports in documented literature. Nonetheless, it is a significant radiological finding that can help provide diagnostic clarity and aid in management, as surgical decompression resulted in partial improvement of neurologic function in one case, albeit with the persistent enhancement of the cord, which has been previously observed.Nava Raj Sharma: Conceptualization; writing – review and editing. Bharosa Sharma: Conceptualization; writing – original draft. SARAL LAMICHHANE: Writing – original draft; writing – review and editing. Madalasa Pokhrel: Supervision; writing – review and editing. Prajwal Shrestha: Resources; writing – review and editing.No funding received.Participating authors have no interests to disclose.None.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Cardiac echinococcosis is a potentially fatal form of hydatid disease; yet, its diagnosis and treatment are challenging due to the variability in its clinical manifestations and due to its various unpredictable preoperative complications. Multi-modality imaging is shown to provide important guidance for the treatment and decision-making. We report a rare case of a 50-year-old woman who had concomitant cardiac and hepatic hydatid cysts. She presented with abdominal pain and elevated eosinophilic white blood cells. The initial abdominal ultrasound and computerized tomography revealed a large cyst in the liver. An intramyocardial cyst was detected by two-dimensional echocardiography. Three-dimensional echocardiography increased the confidence level of two-dimensional echocardiography by displaying the three-dimensional volume of the cyst and allowing visualization of its spatial characteristics and the relationships with adjacent cardiac structures, which was subsequently confirmed at surgery. Multi-detector computed tomography and magnetic resonance imaging helped localize and define the typical morphological features of the cyst. Serology and antigen detection were used for diagnosis. This rare case underlines the integration of clinical, multi-modality imaging, and pathological data in the diagnosis of concomitant intramyocardial and hepatic hydatid cysts. Surgical resection of cysts and anthelmintic medication were successful in the management of this patient. Echinococcus. When present, hydatid involvement often manifests as cystic lesions in many organs  with sinus rhythm at 76 beats per minute and 24-h ambulatory ECG were normal. A chest X-ray examination showed a normal cardiothoracic index and clear lung fields. The patient had eosinophilia (eosinophilic white blood cell 22.1%) in association with a mild increase in plasma liver enzymes  and the erythrocyte sedimentation rate (1h/2h:30/36). Other blood and urine test results were normal.Abdominal ultrasound demonstrated an echo-lucent lesion measuring 150 × 80 mm in the right lobe of the liver. An abdominal computed tomography scan with contrast was performed to assess the tissue characteristics, size, and evidence of local complications and detect other visceral cysts. The cyst was described as an encapsulated structure measuring 115 × 86 mm, with a thin capsule containing homogeneous liquid. The biliary tract was normal on both abdominal ultrasound and CT scan. Cerebral and pulmonary cysts were ruled out by head and chest CT scans, respectively.Transthoracic echocardiography (TTE) revealed a drop-like and echo-lucent intramural structure measuring 29 × 21 mm, bulging into the left ventricle from the myocardium of the left ventricular lateral wall and moving synchronously with the cardiac cycle . On DoppThree-dimensional transthoracic echocardiography 3D-TTE) was performed to image the cyst in multiple planes. In total three different modalities of 3D echocardiography, including live 3D (narrow-angle), 3D zoom, and full volume (wide angle), were acquired and then cropped to visualize the cyst using “en-face” views. The cyst was round and unilocular, with liquid content and well-defined edges; its wall looked homogeneously smooth and hyper-echogenic when seen from outside and inside -TTE was –3. CystiOn cardiac CT images, the lesion looked primarily intracavitary. Contrast-enhanced MDCT of the heart showed an encapsulated, rounded and fluid-attenuated, non-calcified structure along the LV lateral wall measuring 32 mm × 24 mm, its morphologic features looked similar to that of the hepatic cyst which was shown in the 2, respectively. The flow void on CMR confirmed the normal activity of the cardiac valves without intracardiac obstruction.Cardiac MRI also demonstrated a single intramuscular cystic mass located at the anterolateral papillary muscle attaching to LV lateral free wall and protruding into the ventricular cavity . The cysEntamoeba histolytica, Strongyloides, Toxocara, and Echinococcus and examined stool for the eggs of Fasciola hepatica. All results were negative. Because the hepatic cyst was very large and appeared to be at risk for rupture, surgical removal was attempted. However, only a partial cystectomy was possible because it was located too deeply in the liver tissue. The experienced surgeons resected the tip of the cyst and drained all the fluid inside. Echinococcus larvae were found in the histological specimen of the hepatic cyst wall  tests for yst wall ,B. She wEchinococcus granulosus, according to the designation of the World Health Organization and the World Organization for Animal Health ..EchinocoAfter the cardiac cystectomy, the eosinophilic count decreased significantly (13.1% at 4 days post-operation). Post-operative TTE showed an LV cavity devoid of masses, normal LV systolic function, and trivial mitral regurgitation. The patient continued receiving albendazole 200 mg b.i.d for 2 more weeks. Post-operative 12-lead and 24-h ambulatory ECGs were normal. At 1, 6, and 12 months after cardiac surgery, the patient felt well-without symptoms, and her blood tests, abdominal ultrasound, and echocardiographic LV and RV systolic functions were normal. However, the 12-month 2D-TTE revealed mild to moderate mitral regurgitation due to prolapsed A1 and A2 segments that were confirmed by 3D transesophageal echocardiography (3D-TEE) –D; the pEchinococcus granulosus, is common in pastoral areas. In Southeast Asian countries such as Thailand, Vietnam, and Indonesia, Echinococcus is very rare . They become infected by ingestion of eggs passed in dog feces, and this leads to the development of hydatid cysts. During primary infection, eggs may reach the heart through the lymphatic system or via the coronary circulation. Alternatively, cysts in the liver and lungs may rupture and release daughter cysts, larvae, and fragments of the germinal layer, which then settle in the myocardium to produce new cysts. The coronary artery anatomy explains the cardiac distribution of cysts; the most common cardiac location is the left ventricular wall (60%) because the left ventricle receives the largest amount of the coronary blood supply, followed by the right ventricle (10%), interventricular septum (4%), left atrium and left atrial appendage (6%), pulmonary artery (6%), pericardium (7%), and right atrium (3%) (ium (3%) . Cysts mentricle 0%, interium (3%) . Patientium (3%) –17.Electrocardiography may reveal Q-wave or inverted T-wave in leads overlying the affected myocardium or may disclose evidence of conduction abnormalities , 13.Echinococcus in endemic areas using portable ultrasound is considered the best method for early diagnosis, especially in asymptomatic cases  for the publication of any potentially identifiable images or data included in this article.HN, VP, and JK devised the manuscript concept. HN, VP, HP, WT, and HD belonged to the patient's management team. HN and VP performed two-dimensional/three-dimensional echocardiography. HD was the main surgeon. VP contributed to clinical, imaging, and pathological data collection. HN and VP wrote the manuscript in collaboration with JK, WT, HP, and HD. JK and WT reviewed and edited the manuscript. HN took care of revising and submitting the manuscript. All authors read and approved the final version of the manuscript.


Myopericytoma is a rare tumor that arises from perivascular myoid cells. Intravascular myopericytoma is an exceptionally rare subtype with a small number of cases reported. Here, we describe the case of a 31-year-old woman who presented with a lump on the dorsum of the right foot for nine months. Imaging indicated that the lesion is in close proximity to the dorsalis pedis vessels. Following surgical excision, the histological analysis revealed a benign neoplasm arising within a vein wall with features of vascular and pericytic differentiation. When using immunohistochemistry, the blood vessels were highlighted by the cluster of differentiation (CD) 31 and smooth muscle actin (SMA) with negative staining for pancytokeratins. These features led to the diagnosis of intravascular myopericytoma. Myopericytoma (MPC) is considered a soft tissue tumor with a benign nature and was recently discovered . The WorIntravascular myopericytoma (IVMP), first described by McMenamin and Calonje in 2002 , is a unA 31-year-old woman presented with a nine-month history of a lump on the dorsum of her right foot. It started small and gradually increased in size with pain and discomfort. Her medical history had no related information or any evidence of prior trauma. Clinical examination revealed a 2 cm cystic mobile swelling with negative Tinnel's sign.An ultrasound (US) scan showed a heterogeneous, well-circumscribed mass of 19 x 5 x 4 mm of uncertain etiology Figure . AdditioThe patient was discussed in the sarcoma multidisciplinary team (MDT) and the outcome was a mass adjacent to the dorsalis pedis artery with no sinister features. The lesion was completely excised and sent for histology.Macroscopically, it appeared as a firm pale grey tissue measuring 22 x 15 x 7.0 mm. Microscopically, the section showed a well-circumscribed nodular lesion composed of small-caliber thin-walled blood vessels intermixed with a round to oval monomorphic cells, the stroma is fibrous with keloidal changes, and focal thrombosis is noted in the center of the lesion. By immunohistochemistry, the blood vessels were highlighted by cluster of differentiation (CD) 31 and smooth muscle actin (SMA), which were also positive in single scattered cells between the blood vessels, and there was negative staining for pancytokeratins. These findings were in keeping with a benign neoplasm arising within a vein wall with features of vascular and pericytic differentiation and led to the conclusion that the patient had intravascular myopericytoma.An MPC is a benign tumor characterized by a myoid/pericytic line of differentiation. Myopericytes were first described in a report by Dictor et al. in 1992 . The repIt has been observed that a typical MPC consists of monomorphic myoid-like cells with an oval to spindle configuration that have conspicuous multi-layered concentric proliferation around lesion blood vessels . The proIn our case, neuroma was the first differential diagnosis due to the painful nature and position of the swelling, perivascular myoma, angioleiomyoma, angiosarcoma, and hemangiopericytoma were all considered because of the proximity of the lesion to the dorsalis pedis on imaging. Other reactive or neoplastic lesions that might be intravascular should be considered as part of the differential diagnosis for IVMP such as glomus tumor , which lImaging is typically ineffective for evaluating the vascular nature of IVMP. However, US and MRI can be utilized to characterize the morphology and for surgical planning .In summary, we have reported a case of intravascular myopericytoma in the dorsum of the foot. Intravascular myopericytoma is a benign, rare, recently discovered soft-tissue tumor that grows inside blood vessels. Furthermore, the similarity of IVMP to other intravascular lesions makes immunohistochemistry tests essential.