Solitary plasmacytoma is an uncommon hematologic malignancy characterized by the monoclonal proliferation of abnormal plasma cells in the bone or extramedullary tissues and the absence of other multiple myeloma-defining clinical characteristics. Mostly, solitary extramedullary plasmacytoma (SEP) occurs in the head and neck region, also called solitary extramedullary plasmacytoma of the head and neck (SEPHN). Although the standard of care for SEPHN is not well established, either a surgical approach or localized external beam radiotherapy (EBRT) can be used as a definitive treatment. Due to the high radiosensitivity of SEPHN, EBRT has been associated with adequate therapeutic effects in the management of SEP, with the advantage of being a noninvasive modality that yields high rates of local control with a reasonable toxicity profile. We present a case series of three patients with SEPHN treated with EBRT at our institution with clinical outcomes. Solitary plasmacytoma is part of the spectrum of plasma cell neoplasms. It presents as a single mass of monoclonal plasma cells located in bone (medullary), or soft tissues (extramedullary), not in contact with the bone , in the Solitary extramedullary plasmacytoma (SEP) represents 3% of all plasma cell neoplasms and <1% of head and neck tumors .Eighty percent of SEP appears in the head and neck region . The mosCase #1A 58-year-old male with 25-pack-year smoking and without any family history of cancer and a singer presented to an otolaryngology clinic with a five-month history of hoarseness. He denied any additional head and neck symptoms.An office evaluation with laryngoscopy revealed a 2 cm ulcerated lesion in the left true vocal cord with normal mobility. A biopsy from this lesion revealed an ulcerated plasmacytoma comprising kappa-positive and lambda-negative cells. A diagnostic workup for multiple myeloma with bone marrow biopsy, Bence-Jones proteins in urine, bone imaging studies, and serum electrolytes ruled out multiple myeloma and confirmed the diagnosis of solitary plasmacytoma of the larynx.The patient was referred to the radiation oncology department and treated with EBRT to a dose of 40 Gy in 20 fractions over four weeks, using a three-dimensional conformal radiation therapy (3DCRT) technique, with opposed lateral portals Figure . The cliAt 12- and 24-month follow-up visits, laryngoscopy was performed, maintaining a complete clinical response, and subglottic stenosis of approximately 50% was reported.Case #2A 50-year-old female with a family history of her mother with breast cancer and a sibling with unspecified leukemia presented in March 2020 with a six-month history of odynophagia and dysphagia to solids.A computed tomography scan showed several solid lesions in the left base of the tongue extending to the floor of the mouth, and the largest lesion measured 19 mm × 16 mm × 20 mm. No enlarged cervical lymph nodes were found  and image-guided radiation therapy (IGRT) Figure . The groGrade 1 xerostomia was noted at three months after EBRT without any late side effects. A computed tomography scan at six months revealed no evidence of tumoral activity Figure . A repeaCase #3A 56-year-old male with no relevant medical history presented with repetitive episodes of epistaxis, requiring nasal packing in the emergency department. A nasal endoscopy was conducted in the emergency room, observing profuse active bleeding, impending observing the source of the bleeding, and limiting the procedure to hemostasis only. Computed tomography showed a soft tissue mass in the left nasopharynx occupying the entire space, thereby obstructing both choanae and partially extending into the nasal cavity with homogeneous enhancement upon contrast administration  or when an organ-sparing treatment is desirable as in tumors originating in the larynx (case #1).Given the high radiosensitivity of SEPHN, radical surgery should be avoided as primary treatment as it can be mutilating ,2, thereEBRT dose for SEPHN is not well-established, with doses ranging from 30 to 60 Gy in 1.8 to 2 Gy per fraction -9. EBRT Dose/fractionations recommendations are based on the tumor size, with 40 Gy being prescribed for tumors smaller than 5 cm and 50 Gy for tumors larger than 5 cm . NonetheThe EBRT treatment volume comprises the GTV, which includes the observed lesion in computed tomography simulation with or without magnetic resonance imaging. The CTV consists of the GTV plus a 0.5 to 1 cm expansion respecting anatomic boundaries. A PTV to account for daily setup uncertainties during treatment is subsequently created by adding a 0.5 to 1 cm margin ,17,18. WRegarding EBRT techniques, the treatment volume and critical structures may largely dictate the choice of treatment modality for each case. Conformal EBRT with parallel fields is commonly used. However, as previously stated, earlier studies were published before IMRT/VMAT techniques were widely implemented worldwide. Hence, in some situations, more conformal techniques such as IMRT and VMAT may be the preferred option to spare critical structures, minimizing toxicity while maintaining therapeutic dosage, usually at the cost of a larger total volume of normal tissue irradiated, but to a lower dose. IGRT delivery may offer a clinically appropriate advantage, particularly for treatment sites that are adjacent to critical dose-limiting normal structures ,17.In the present case series, we report the clinical presentation and characteristics of radiotherapy, toxicity, and clinical outcomes of three patients with SEPHN. In case #1, the patient remains disease-free at two-ya ear follow-up but with long-term toxicity presented as subglottic stenosis. Although cases #2 and #3 require longer follow-ups, we believe that the prescribed treatment will provide adequate local control of the disease, as has been described in larger case series ,9,13,16.Similarly, we believe that the use of a more modern EBRT technique such as VMAT as in cases #2 and #3 can help reduce long-term side effects, unlike case #1, who was treated at our institution when advanced EBRT techniques were not yet available.Although patients with SEP show a lower rate of progression to multiple myeloma than patients with solitary bone plasmacytoma (25%-35% vs. 65%-84% at 10 years) .SEPHN is an uncommon hematologic malignancy in which the standard of care is not well defined. Nonetheless, it is crucial to understand its high radiosensitivity and biological behavior as, in carefully selected cases, EBRT (3DCRT and IMRT/VMAT) has been associated with excellent control rates. As presented in our case series, EBRT is a well-tolerated treatment with an acceptable toxicity profile providing adequate control of the disease with the dose ranging from 40 to 50 Gy in conventional fractionation.Moreover, normal tissue sparing and fair aesthetic and functional outcomes are paramount, especially in head and neck subsites. There a surgical resection could be mutilating and can compromise organ functioning as in SEPHN originating in the larynx and oropharynx (cases #1 and #2) or anatomical sites where the tumor is not easily accessible for surgeons or as in tumors affecting the nasopharynx (case #3), making EBRT the treatment of choice.


Vulvar and vaginal lesions representing a wide spectrum of diseases in female lower genital tract diseases make up a small part of all gynecological etiologies. Many of them are rare etiologies and are reported in case-reports studies. Translabial and transperineal ultrasound are modalities of choice for the first evaluation of perineal lesions. MRI is usually performed to determine the etiology of the lesions and stage. Benign lesions of the vulva and vagina usually manifest as simple cystic (vestibular cyst or endometrioma) or solid lesions (leiomyoma or angiofibroblastoma), while malignancies usually appear as large, solid masses and fill into both vaginal and perineal area. Post-contrast images play an important role in establishing a differential diagnosis, however, some benign lesions can also exhibit a vivid enhancement. Knowledge about radiologic-associated pathological manifestations may aid clinicians in better understanding these pathologies, especially for some rare lesions, and making a proper diagnosis before invasive procedures. Vulvar cancer is the fourth most common gynecologic malignancy. Both translabial and transperineal ultrasound are modalities of choice for the first evaluation of perineal lesions. Compared with sonography, MRI has better soft tissue resolution and is more commonly used to outline, diagnose, and stage these conditions in clinical units. Unlike lesions in the cervix and uterus, some tiny lesions in the perineum may be overlooked even on multiplanar MRI. Thus, scrutinized review of pre-operative MRI plays a vital role in subsequent treatment design. The final diagnosis depends mainly on histological findings. This article reviews the MRI manifestations of various abnormalities of the vulva and vagina. Furthermore, the correlation of MRI with pathological findings is also comprehensively reviewed, providing its advantage in the differential diagnosis between MRI and pathological findings.Female lower genital tract diseases, mainly including vulvar and vaginal abnormalities, represent a wide spectrum of diseases (T1WI/T2WI with or without fat-saturation (FS) technique/DWI in either axial, sagittal, or coronal scan plane) and some complicated sequences (e.g. high resolution or dynamic contrast-enhanced scan) based on various suspected etiologies (SupplementaryTable 1-4). Contrast material is routinely used for both vulvar and vaginal lesions during MR scans, especially for malignant lesions , the longer acquisition time will increase the movement-related artifacts and decrease the image quality. In our institution, both equipments are applied for clinical purposes. Scanning sequences will cover conventional protocols  affects approximately 10% of females of reproductive age. Endometriosis can occur in the uterus  or some other anatomic structures.is found . PolypoiT1WI, representing bleeding components. The FS-T1WI sequence is especially important because ES always shows a high signal, which is helpful to differentiate ES from other lesions with a pure high signal on T1WI. The T2-shading effect, sometimes, may not be obvious owing to small lesions displaying a homogeneously high signal .Macroscopic appearance shows that it is lobulated, well-circumscribed, and sometimes covered by a thin capsule. The cut surface is homogeneously yellow, with fibrous septa dividing into lobules. The tumor consists of a relatively uniform population of mature adipocytes arranged in sheets and/or separated into lobules by thin fibrous septa. Adipocytes and septal stromal cells lack cytological atypia. Moreover, lip blasts are not present  is a rare tumor. When it was first reported in 1999, it was considered a unique etiology differentiated from other mesenchymal tumors of the skin. Tumor cells consistently express estrogen receptors and progesterone receptors. Finally, the chance of recurrence is low.Some pathological features are listed below. First, the tumor is well demarcated with a tan-white and solid cut surface. Second, the diameter of AFB is usually less than 5 cm. Third, it has alternating zones of cellularity and prominent small to medium-sized thin-walled vessels in the edematous matrix, which are clustered adjacent to blood vessels in the loose edematous matrix. The morphology of tumor cells is spindled to ovoid, with a centrally to eccentrically located round nucleus, giving them a plasmacytoid appearance, mitotic activity is too rare to be identified. Some quantities of stromal collagen fibrils or sclerotic plaques are present . Fourth,T1WI and hyperintensity signal on T2WI, while the tumor homogeneously enhanced similar to the normal uterus on post-contrast images . The differential diagnosis of LM is usually with leiomyosarcoma (LMS).The gross and microscopic features are similar to their uterine counterparts. They are usually submucosal and composed of spindle cells or occasionally epithelioid cells.T2WI. LM usually displays as a well-circumscribed mass with a homogeneously intermediate signal on both T1WI and T2WI. If some degeneration within the tumor occurs, then the foci of high signal on T2WI are also seen. On DWI, the tumor can show a mildly restricted signal. On contrast-enhanced MRI, the mass often shows obvious enhancement, which can be mistaken for malignancies .Other entities might include Bartholin’s cyst, periurethral cysts, abscess, hematoma, lichen sclerosis, nerve sheath tumors, and so on for the vulva and perineum alone. Most of these lesions are accidentally found during complaints of other severe complications at the clinic. Most of the patients with benign etiologies will undergo conservative therapy and we cannot compare some connections. Among them, Bartholin’s cyst is the most common benign cyst in the major labium with a pure high signal on  Symptoms may include perineal pruritus, pain, a lump, or an ulcer. SCCs of the vulva account for 90% of vulvar cancers and 5% of gynecologic cancers.Vulvar cancer is an uncommon gynecologic malignancy primarily affecting post-menopausal females. Squamous cell carcinoma (SCC) is the most common subtype among patients with vulvar cancer. The tumor cells typically show strong diffuse p16 staining (due to HR-HPV integration), but usually, wild-type p53 expression. A p53-associated pathway in approximately 40% of cases, which usually occurs among females in the seventh to eighth decades of life, leads to keratinizing SCCs. It is usually not related to HPV but associated with one or more of the causes such as differentiated vulvar intraepithelial neoplasia, lichen sclerosis, squamous cell hyperplasia, and p53 mutations . The boundary between the tumor and adjacent structures  will help to precisely stage pre-operatively. In general, the suspected nodes in the pelvis with more than 10 mm in the short diameter now are considered as the cut-off value for potential malignant nodes. According to the newest FIGO stage, if any inguinofemoral node with more than 5 mm in diameter, then the tumor will be divided into FIGO Stage III or more.The tumors often appear as the focal thickness of the skin. The large tumor usually appears as soft tissue mass signals with homogeneous or heterogeneous enhancement after contrast agent injection . Vulvar  abdomen .2,3,28 It may present as a focal/multifocal lesion, which is difficult to be differentiated from erythema and eczema clinically. PD, accounting for approximately 1% of vulvar cancers, occurs in the late reproductive and post-menopausal age groups, with a median and mean age in the seventh decade. Intraepidermal recurrence occurs in up to 60% of cases due to incomplete excision. In most studies, the risk of recurrence does not correlate with resection margin status. Rarely, there is a progression to invasive PD during the follow-up period.Extramammary PD is a rare etiology that usually develops in apocrine gland-rich areas, such as the vulva, scrotum, and penis.in-situ adenocarcinoma of the vulvar skin, with or without underlying invasion. Secondary involvement of the vulvar skin by carcinoma of rectal, bladder, or cervical origin is designated “secondary PD”. Vulvar PD is thought to originate from pluripotent stem cells in the epidermis or skin appendages, arising from either the labium majus or labium minus and can extend to the extravulvar skin and/or involve the vaginal or cervical mucosa. PD typically presents as mild eminence and redness of the skin with ill-defined margins, compared with the adjacent normal skin. Paget cells are diffusely and strongly positive for CK7  refers to gestational trophoblastic lesions that have the potential for local invasion and metastasis. The reported incidence varies between 1/600 in therapeutic abortions and 1/1000–1200 in normal pregnancies. Ultrasound is the first-choice modality in diagnosing GTN. Combined with an elevated hCG level for normal gestational age is highly suggestive of molar pregnancy or GTN. The molar tissues may show a “grape appearance” in both the cavity and myometrium. If solid components consistently persist, then the pre-operative diagnosis should be cautiously diagnosed and needs to be differentiated from an incomplete miscarriage or an ectopic pregnancy as vivid enhancement may be seen in all these abnormal etiologies. For post-treatment evaluation of GTN on MRI, the multicystic lesions with hemorrhagic components displaying as a high signal may present as the primary manifestation. The enlarged vessels surrounding the lesion may appear as flow void signals in every protocol of MRI. The hemorrhagic components often show a high signal on T1WI. MR angiography or venography helps image potential coexistent vascular malformations . These lesions together appear as the blood pool on maximum intensity projection reconstructed images .MRI is not routinely used in clinics to image GTN, mostly dedicated for GTN staging and evaluation of complex GTN. The common metastatic sites are the lungs (80%), vagina (30%), pelvis (20%), liver (10%), and brain (10%). LMS includes three subtypes: spindled, epithelioid, and myxoid. There is a solitary mass, usually with a diameter of more than 5 cm, and circumscribed or infiltrative borders. The cut surface is soft or firm and fleshy white-tan-gray with variable hemorrhage, necrosis, and cystic change. Classically, the criteria for malignancy are at least two of the three features: moderate to severe cytological atypia, mitotic count >10 mitoses/10 HPF of 0.53 mm in diameter and 0.22 mm2 in area, and tumor cell necrosis ; young adults and post-menopausal females may also be affected. The tumors present as vaginal bleeding and vaginal mass which is typically soft, edematous, nodular, papillary, or polypoid (sarcoma botryoides), often with introital protrusion.Rhabdomyosarcomas (RMS) is the most common vulvar malignancy in pediatric patients (median age of onset is 16-years-old).There is a densely cellular zone (cambium layer) of primitive, small, mitotic cells under the squamous epithelium that may be invaded by tumor cells. Beneath the cambium layer is a hypocellular edematous zone of similar small cells and rhabdomyoblasts; foci of hyaline cartilage may also be seen. The rhabdomyoblasts, which may be sparse, vary from round to strap-shaped. They have eosinophilic cytoplasm with cross-striations in most cases. Immunoreactivity of desmin and more specifically, skeletal muscle markers  facilitate the diagnosis . The tumT1WI and T2WI, which is confirmed by histological findings. Heterogeneous enhancement is often observed on MRI of RMS . RMS could involve the cervix, vagina, and vulva. They generally appear as cystic-solid masses with isointense signals on both I of RMS , in whic Compared with cutaneous melanoma, the prognosis for VM is still worse . Primary VM is also an extremely rare tumor of the lower genital tract, accounting for only 0.3–0.8% of all melanomas in females. Malignant melanoma is a malignant melanocytic neoplasm arising in a non-cutaneous site of the female genital tract, usually in the vulva or vagina  and rarely in the cervix. Malignant melanoma accounts for 5% of vulvar and vaginal malignancies and is most common in older females with a mean onset age of 65 years.Vulvar melanoma (VM) accounts for 1% of all melanomas in females and 5% of all vulvar malignancies. They usually present as large, ulcerated, and nodular masses; Moreover, they often involve the cervix. The tumor is usually formed by sheets or expansive nodules of large pleomorphic epithelioid or (less commonly) spindle malignant melanocytic cells. Melanin production is variable but usually focally present within melanoma cells. Immunoreactivity of S100, SOX10, HMB45, and Melan-A (MART1) facilitates the diagnosis . If vulvar lesions are small, they need to be differentiated from SCC owing to the overlapping imaging signs . To be noted, even if no high signals on T1WI, VM is still not definitely excluded. Post-contrast images may provide limited information in differentiating between LM and LMS because both trilogies enhance moderately or vividly. RMS is the most common vulvar malignancy in pediatric patients with the characteristic feature: a polypoid botryoid mass on post-contrast MRI. Clinical signs and symptoms are still necessary to establish a proper diagnosis. The final diagnosis is based on histological findings. To reasonably utilize an MRI scan pre-operatively will aid in decision-making and individual treatment plan.In summary, vulvar and vaginal lesions cover a wide spectrum of etiologies with mimicking imaging findings. Owing to the high soft tissue resolution, MRI helps delineate the extent of the primary lesion , determine the etiology, and evaluate the metastatic lesions. Superficial lesions cannot be satisfactorily displayed on MRI and the role of MRI is to determine the extent of lesions and coexistent lesions. Endometrial cysts in the lower genital tract and peritoneal area should be included as the differential diagnosis other than the most common lesions . For solid masses in these areas, LM is the common benign etiology with a clear margin; while SCC is the most common malignant tumour, in which MRI has a critical role in staging. LMS and VM are also needed to be considered when having a large mass with an irregular margin (LMS) and high signal on plain Knowledge about vulvar and vaginal pathology will help establish a proper pre-operative MRI diagnosis.MRI plays an important role in staging malignant tumors located in the lower genital tract.The diagnosis of small and tiny lesions developing in the perineum relies on histological findings thereafter.


Whole-exome sequencing showed that this tumor also harbored CDK4, TSPAN31, and JUN amplification. At the latest follow-up (85 months after surgery), the patient was alive, with no evidence of disease. To the best of our knowledge, this is the first example of a molecularly confirmed primary bladder liposarcoma and the first case of DDL at this site.Liposarcomas originating in the urinary bladder are extremely rare. Only six cases of bladder liposarcoma have been reported, and all have been described as myxoid liposarcomas. Notably, none of the patients underwent molecular testing. Here, we report a dedifferentiated liposarcoma (DDL) that occurred in the urinary bladder, primarily in a 69-year-old Chinese woman, with infrequent low-grade dedifferentiation. Computed tomography (CT) revealed an ill-defined solid mass in the anterior bladder wall. The patient underwent a partial bladder resection. Histologically, the tumor cells with mild-to-moderate nuclear atypia were arranged in fascicular and storiform patterns, mimicking a low-grade fibroblastic tumor. In addition, scattered small foci of typical lipoma-like well-differentiated components were identified. Immunohistochemically, the tumor tested positivity for MDM2, CDK4, and p16. Fluorescence  In particular, MDM2 is amplified in almost all cases of ALT/WDL/DDL, and detection of MDM2 amplification by fluorescence in situ hybridization (FISH) has been recognized as the gold standard for the diagnosis of this tumor  among adults, accounting for approximately 15%–20% of all STSs  and cut into 4-μm sections for examination. Standard immunohistochemical staining was performed using the EnVision Plus detection system  with positive and negative controls. Information on the antibodies is presented in 2.2MDM2 Spectrum Orange Probe , GSP CDK4 Gene Amplification Probe and FRS2 Probe  were used for MDM2, CDK4, and FRS2 amplification. ALK rearrangement was performed using the Vysis LSI ALK Dual Color Break Apart Rearrangement Probe . FISH tests were performed according to an established protocol  detection were CTNNB1-F: 5’TCCAATCTACTAATGCTAATACTGTTTCGTA3’ and CTNNB1-R: 5’ TCCAATCTACTAATGCTAATACTGTTTCGTA3’. Sanger sequencing was performed by BGI Genomics Co., Ltd. .The primers for 2.4WES was performed by Novogene Technology Co., Ltd.  on FFPE tissues to explore possible molecular abnormalities.3A 69-year-old female was admitted to our hospital with a one-month history of recurrent gross hematuria, accompanied by slight urgency and pain during urination. The patient had no history of trauma or infection. Physical examination revealed an approximately 5 cm × 4 cm palpable fixed smooth mass in the hypogastrium with no tenderness or rebound tenderness. Urine cytology revealed an increased number of leukocytes with no heterotypic cells or red blood cells. Computed tomography showed an ill-defined solid mass in the anterior wall of the bladder Figure 1Macroscopic examination revealed a partially excised bladder measuring 7 cm × 7 cm × 5 cm. The overlying urothelium is rough. Thickening of the bladder wall was almost completely replaced by gray-white woven lesions, which showed diffuse infiltrative growth into perivesical soft tissues. On the cut surface, a gray-white and tough mass resembled the scar tissue.Microscopically, the urothelium and lamina propria of the bladder showed no abnormalities  and urine tests 1 month after the operation, every 3–6 months for 3 years, and then annually. During an 85-month follow-up period, the patient was alive with no evidence of disease recurrence.4This study describes a case of primary liposarcoma arising from the urinary bladder. Bladder mesenchymal tumors are rare, representing 1%–5% of all primary urinary bladder tumors, and leiomyomas and leiomyosarcomas are the most common benign and malignant mesenchymal tumors of the bladder, accounting for approximately 0.43% and 1% of all bladder tumors, respectively , 14. AltFUS/EWSR1::DDIT3 fusion gene. In addition, ML occurs predominantly in the deep soft tissues of the extremities  and rarely occurs in other sites, including the retroperitoneum. Previous studies proposed that there was no existence of true primary retroperitoneal ML, and these “retroperitoneal ML” were proven as metastasis or DDL with myxoid-like changes (DDIT3 arrangement in a large series study (All previously reported bladder liposarcomas were ML and round cell liposarcomas (high-grade ML). However, all published bladder ML studies lack genetic confirmation of the  changes , 16. Onles study . Owing tNotably, the current case exhibited peculiar low-grade fibroblastic tumor-like morphological features. DDL has a wide morphological spectrum, and most dedifferentiated areas exhibit an intermediate- to high-grade histological morphology, resembling undifferentiated pleomorphic sarcoma or myxofibrosarcoma. DDLs with low-grade differentiation have increasingly been recognized in recent years, and these dedifferentiated areas are histologically similar to low-grade myofibroblastic sarcoma, fibromatosis, or inflammatory myofibroblastic tumors (IMT) . BesidesFRS2 amplification. FRS2 is located close to MDM2 within the 12q13-15 region and is amplified in >90% of ALT/WDL/DDL and low-grade osteosarcoma cases (MDM2(+)/FRS2(−) ALT/WDL/DDL . All three previously reported MDM2(+)/FRS2(−) DDLs were in peripheral sites, and their dedifferentiation components included two homologous pleomorphic liposarcoma-like and one intermediate-grade fibrosarcoma-like. The present case represents the first case of MDM2(+)/FRS2(−) DDL located in a central site with a low-grade dedifferentiated area. Furthermore, amplification of the JUN gene was identified in this lesion. Outside of 12q13-15, amplification of 1p32 (including JUN) and 6q23 (including ASK1) was observed in both ALT/WDL and DDL but was more correlated with undifferentiated histology /FRS2(−) genotype in this case has been reported to be associated with indolent clinical behavior  for the publication of any potentially identifiable images or data included in this article. Written informed consent was obtained from the participant/patient(s) for the publication of this case report. Written informed consent was obtained from the participant/patient(s) for the publication of this case report.JC and RP analyzed the data and prepared the manuscript. YZ collected the clinicopathological data of the patient. YL and MC helped with molecular experiments. XH helped histopathology review. HZ was responsible for the diagnosis, study design and the manuscript revision. All authors contributed to the article and approved the submitted version.


A female newborn weighing 542 g and delivered at 27 weeks gestation presented with bilateral inguinal hernias while in the neonatal intensive care unit. Ultrasonography confirmed herniation of the uterus into the right inguinal hernia without signs of incarceration. Due to the absence of complications, she was discharged and scheduled for follow-up at the outpatient clinic. At 11 months of age, a subsequent ultrasonography showed only omental herniation, with no evidence of uterine prolapse. When she reached 1 year of age, a laparoscopic percutaneous extraperitoneal closure procedure was performed. During the surgery, it was observed that the uterus and fallopian tubes were located near the hernia orifice, but no clear prolapse was detected. The procedure concluded safely with successful high ligation. It has noted that in cases of uterine prolapse hernias, the uterus tends to recede as the child grows, which supports the decision to delay surgery for improved safety. The occurrence of inguinal hernias in childhood ranges from 0.8% to 4% . ApproxiA female newborn weighing 542 g was delivered via emergency cesarean section at 27 weeks of gestation due to concerns about the fetus’s well-being. Due to her extremely low birth weight, she was admitted to the neonatal intensive care unit (NICU) and required respiratory and circulatory support immediately after birth. Chromosomal analysis using G-banding indicated a normal karyotype of 46, XX without any observable abnormalities. As the infant’s condition stabilized and she gained weight, she was discharged from the NICU on postnatal day 183, weighing 2928 g.During the newborn’s stay in the NICU, both inguinal regions showed swelling. An ultrasound examination conducted when the infant was 3 months old revealed bilateral inguinal hernias, with the uterus herniating on the right side . There wWhen the patient turned 1 year old, a laparoscopic percutaneous extraperitoneal closure (LPEC) procedure was carried out to address the bilateral inguinal hernias. Although the uterus was slightly deviated toward the right side of the pelvis, its position appeared to make it difficult for the uterus to herniate through the hernia orifice. The right fallopian tube was located near the hernia orifice, and a high ligation was carefully performed to avoid entanglement of the fallopian tube . The proWe encountered a case involving a uterus-containing inguinal hernia in an infant and observed its natural progression. As the child matured, the prolapse of the uterus diminished. This resolution of the hernia was believed to be due to the improved positioning of the uterus toward the previously herniated side, which had exhibited a tilt, as a result of the child’s aging and growth .Sliding hernias involve the displacement of various organs, including the ipsilateral ovary, fallopian tube, and even the uterus. The inguinal canal has the capacity to accommodate different intra-abdominal organs, such as the omentum, small intestine, appendix, Meckel’s diverticulum, ovary, fallopian tubes, and bladder. In female infants, sliding inguinal hernias can occur, affecting the ipsilateral ovary and fallopian tube, and there have been reported cases involving both ovaries. It is important to note that the uterus can also become trapped within these hernias , 4. The The presence of the uterus within the hernial sac is typically observed in individuals who are hermaphrodites or intersexual variants or have a male phenotype 46, XY). Male pseudo-hermaphroditism, which involves the presence of Müllerian duct derivatives such as the uterus, cervix, fallopian tubes, and upper one-third of the vagina, can occur in phenotypic males . In chil6, XY. MaInguinal hernia repair is a commonly performed surgical procedure in pediatric patients, and the standard approach involves high ligation of the hernial sac . HoweverUterus-containing inguinal hernia is an extremely rare, and ultrasound examination is useful for its diagnosis. When there is no incarceration, the presence of uterine prolapse alone does not require immediate surgical intervention. We consider that waiting for growth would resolve the uterine prolapse, leading to a higher likelihood of performing a safer surgical procedure.The authors declare no conflict of interest.None declared.


Multiple hemorrhagic brain lesions are mainly diagnosed based on clinico-radiological features integrated with histological data. Intravascular papillary endothelial hyperplasia (IPEH), or Masson's tumor, is a very rare entity, particularly when localized in the brain. In this study, we describe a case of multiple recurrent brain IPEHs and provide details on the diagnostic phase, therapeutic approaches, and related challenges. A 55-year-old woman presented with a relapsing neurological deficit. Brain magnetic resonance imaging (MRI) revealed a hemorrhagic right frontal-parietal lesion. When new neurological symptoms occurred, subsequent MRI scans detected more bleeding cerebral lesions. She underwent a series of single hemorrhagic lesion debulking. For any samples that underwent histopathological examination, the first results were not informative; the second and the third results revealed hemangioendothelioma (HE); and the fourth results led to the IPEH diagnosis. Interferon alpha (IFN-α) and subsequently sirolimus were prescribed. Both were well tolerated. Clinical and radiological features remained stable 43 months after starting sirolimus therapy and 132 months after the first diagnosis. To date, 45 cases of intracranial IPEH have been reported, mostly as single lesions without parenchymal location. They are usually treated by surgery and sometimes by radiotherapy upon recurrence. Our case is notable for two main reasons: because of the consecutive recurrent multifocal exclusively cerebral lesions and the therapeutic approach we used. Based on multifocal brain recurrence and good performance, we propose pharmacological therapy, including IFN-α and sirolimus, to stabilize IPEH. Multiple hemorrhagic brain lesions are primarily due to hypertension and cerebral amyloid angiopathy. Secondary causes include vascular malformations, coagulopathy, hemorrhagic conversion of ischemic stroke, hemorrhagic neoplastic localizations (metastases or vascular tumors), trauma, vasculitis, stimulant drugs, or sinus venous thrombosis . VasculaClinico-radiological features and histological reports of such hemorrhagic brain lesions are essential to make the diagnosis, but histologically, a definitive diagnosis has proven difficult because the hemorrhages destroy the tissue. Moreover, some vascular tumors may have similar or overlapping morphological features and immunophenotypes.International Society for the Study of Vascular Anomalies (ISSVA) Classification, https://www.issva.org/classification), a rare condition mostly localized in the skin and subcutaneous tissues. Our report is remarkable for noting an exclusively intracranial recurrent multifocal IPEH, stabilized during immunomodulation with interferon-alpha (IFN-α) and mechanistic target of rapamycin (mTOR) inhibitor treatment.We describe a case of intravascular papillary endothelial hyperplasia (IPEH), or Masson's tumor . In particular, she had dysesthesia of the left hand that, within 2 days, extended to the whole arm and half-face, which was associated with dysarthria. The latter symptom disappeared within a few days. She did not complain of having a headache, and the physiological parameters, including body temperature, blood arterial pressure, and heart and respiration rate, were normal. Her past medical and psychosocial history was unremarkable, as well as her family health history. Brain magnetic resonance imaging (MRI) performed 4 days after clinical onset showed a right frontal-parietal hemorrhagic lesion . After 1After 3 months (Time 3), the patient reported headache and aggressive mood that lasted for a few weeks; three new brain lesions  were identified on MRI. The left basal frontal nodule  was exciAfter 4 months of the initial visit (Time 4), the patient presented with acute-onset right arm palsy; a brain MRI revealed a new lesion in the left motor cortex . She proAfter 1 month (Time 5), considering the number of lesions and their evolution, and based on anecdotal cases, treatment with IFN-α (3 million UI × 3/week) was started and continued for 12 months. She showed good tolerability to the treatment and experienced grade 2 leukopenia only and no further complications.The patient was radiologically and clinically stable until the 20th month from the beginning (Time 20) when she presented with acute severe dizziness. MRI revealed a right hemispheric cerebellar mass with radiological features similar to that of the previous lesions . Due to After 80 months of the initial onset (Time 80), the patient presented with acute severe headache, confusion, and memory impairment. Brain MRI revealed a large hemorrhagic left frontal lesion . The pat/eosin and Masson's trichrome staining and immunohistochemical analysis using the abovementioned antibodies.Histopathological examination of the surgical specimens was performed using hematoxylinIn addition, previous histological specimens (obtained at time points 3 and 4) were reviewed; nevertheless, due to the hemorrhagic components, a differential diagnosis between HE and IPEH could not be made using these samples.In two brain MRIs, obtained at the 87th month (Time 87) and 89th month (Time 89) from the onset, new hemosiderin alterations in the right internal subcapsular , left anAt the onset of the disease and 2 years of follow-ups, an extended check-up was done, including complete blood tests, whole-body computed tomography (CT), scintigraphy, and positron emission tomography, and all results were negative.IPEH is characterized by endothelial proliferation strictly linked to thrombosis; however, the specific etiopathology of the disease is still largely unknown. It was first described by Masson in 1923 and had been called IPEH since 1976 .The histological diagnosis of brain-localized IPEH can be challenging mainly due to the destruction of the hemorrhagic component of the lesions, leading to easy misdiagnosis as other conditions. Furthermore, the differential diagnosis includes similar or partially overlapping lesions such as HE, a rare low-proliferation vascular tumor with intermediate behavior between a hemangioma and an angiosarcoma, with possible local aggressivity, occurring in many organs and soft tissues.* images due to hemosiderin. However, some characteristics of other vascular lesions sustained the hierarchy of differential diagnoses. Hemangiomas can be multiple synchronous or asynchronous, can be small, and show hypointensity in T2* w.i. in addition to hemorrhagic events; they often show caput medusae on enhanced T1 w.i. and calcifications on CT (both features absent in our patient); moreover, such lesions very seldom keep on appearing in a short time in older adults. Cerebral angiosarcoma often shows well-demarcated borders, high-flow serpentine vessels  in an otherwise solid non-specific soft-tissue mass, cystic or necrotic areas within the lesion (absent in any of our case report lesions), sometimes intralesional calcifications; enhancement can be moderate or strong but mostly heterogeneous and more pronounced on the margin (unlike the lesions in our patient); perilesional edema is often present; angiosarcoma evolves locally with enlarging lesion (contrary to our case) and tends to disseminate. HE is intermediate between angiosarcoma and benign hemangioma because of its tendency to slowly enlarge and metastasize  tumors while it was considered grade 1 before) within the brain primary hemangioendothelioma and is often a single mass lesion.There are no specific radiological features that characterize IPEH, and the final diagnosis requires careful histological examination. All tumors appear as mildly hypointense to isointense on T1-weighted images, except for those with recent bleeding which are hyperintense; enhancement on T1 contrasted images is mostly observed; lesions with previous or subacute bleedings show a signal loss on T2Regarding histology, hemangiomas are composed of compact vessels arranged in a multilobular configuration, with inconspicuous to dilated luminal spaces, sclerotic stroma, and vascular walls. HE typically shows well-differentiated nests and cords of cells, often within a myxoid matrix, with abundant eosinophilic cytoplasm and small intracytoplasmic lumina. Angiosarcoma presents irregular vascular channels with poorly differentiated areas, composed of cells characterized by high pleomorphism, atypia, and mitotic activity, and causes more parenchymal destruction than HE while causing less sclerosis. Moreover, HE is characterized by the unique CAMTA1-WWTR1 fusion or YAP1-TFE3 fusion.n = 37/45). The prevalence is higher in female patients (F:M = 36:9). All the reports are summarized in n = 18/45: seven frontal, five parietal, two temporal, and four in more than one lobe) and to less extent in the infra-tentorial region , pineal region (n = 2/45), or other location .Until December 2022, 45 cases of intracranial IPEH have been reported in peer-reviewed literature, mostly as single lesions without parenchymal localization. Out of 45 patients, 18 had secondary cavernous malformation or aneurysm/angioma, often following radiotherapy . The medBased on the monoistitutional database, Roach et al.  estimateMost patients underwent surgery as initial treatment, and in a few cases, radiotherapy was performed on recurrence. Unfortunately, most reported cases had a short follow-up .We describe a CNS parenchymal primary IPEH presenting with focal neurological signs, which is reported in <40% of cases with parenchymal brain IPEH. To the best of our knowledge, only one multifocal parenchymal case has been reported. In that case, a woman experienced headache and focal signs and underwent exeresis of one lesion, with no progression of disease at the 6-month follow-up . Others Treatment data displayed in the literature are limited to case report/series, most of which describe single-lesion cases or have a very short follow-up. The treatment of choice for IPEH is surgery, eventually associated with embolization and/or radiotherapy. In two pediatric cases only, systemic therapy has also been reported: Sim et al. described the case of a single para-sellar IPEH treated twice by surgery followed by adjuvant IFN-α, while Sickler et al. reported a case of middle cranial fossa probable IPEH treated with surgery and doxorubicin plus dacarbazine , 16.Our case is noteworthy because of the consecutive multifocal incurrence of exclusively parenchymal brain lesions with a long follow-up (132 months). The approach used to study and the follow-up of the patient were comprehensive, including brain imaging (CT and MRI) as well as total body investigation. We did not proceed with cerebral angiography due to the decision to perform surgical excision of the hemorrhagic symptomatic lesions.We needed to attempt a systemic treatment strategy for the patient to avoid the collateral effects of consecutive repetitive surgeries and eventual large-dose radiotherapy.Due to the initial diagnosis of HE, we opted for IFN-α treatment, according to the literature . PreviouThe disease evolution in our patient subsequently led to the diagnosis of IPEH. Based on this, combined with the recurrent multifocal brain evolution and the good performance, the treatment decision shifted to sirolimus, an old drug repurposed for anti-proliferative action, already experimented for “extensive and/or complex slow-flow vascular malformations” (NCT01811667) . SirolimThe case we described got recurrent surgical exeresis and corresponding histology, associated with dedicated neuroimaging, thus allowing an extensive study; we then approached it with systemic therapy, resulting in success. However, as in any case report, this is not enough to prove the efficacy of IFN-α and sirolimus in such a context, and the data need to be replicated.In the case described herein, sirolimus, which was started after close multifocal recurrences of brain IPEH, enabled disease stabilization over 43 months of ongoing treatment and for 132 months after onset .The patient has tolerated the long-term low-dose sirolimus treatment well.The challenging diagnostic and therapeutic approach reported is anecdotic, but it may be helpful in patients affected by such rare pathological entities.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Ethical review and approval was not required for the study on human participants in accordance with the local legislation and institutional requirements. Written informed consent for participation was not required for this study in accordance with the national legislation and the institutional requirements. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.EA: drafting/revision of the manuscript for content, including medical writing for content, study concept or design, and analysis or interpretation of data. BP and MS: analysis or interpretation of data. PF, DA, GD, and FF: drafting/revision of the manuscript for content and including medical writing for content. LC: study concept or design. VC: drafting/revision of the manuscript for content, including medical writing for content, major role in the acquisition of data, and analysis or interpretation of data. All authors contributed to the article and approved the submitted version.


Medical records of a 76‐year‐old woman with a recurrent acinic cell carcinoma of the left parotid gland with lateral skull base invasion were reviewed. She underwent subtotal petrosectomy followed by radiation therapy. After surgery, she remained disease‐free for more than 16 months. The patient reported onset of symptoms 3 months earlier. Physical examination showed no facial paralysis. Ten years before she had a left breast cancer and actually she took no medications. She was not an active smoker with no alcohol or drug use.A CT of the neck after contrast showed a 2 × 3 × 3 cm heterogeneously enhanced mass in the deep lobe of the left parotid gland, without extraparotid extension. No lymph node abnormalities were observed.An ultrasound guided fine needle aspiration biopsy (FNAB) was performed and the citology suggested a poorly differentiated basal cells neoplasm (cT2N0M0). Consequently, the Head and Neck Tumor Board recommended a total surgical excision.The patient underwent a total left parotidectomy with facial nerve monitoring and facial nerve preservation. She was discharged 5 days after surgery and recovered without incident. A final pathology showed an AciCC of the left parotid gland with low degree of malignancy, without perineural invasion and with free resection margins (pT3N0M0).As a result, the Head and Neck Tumor Board, supported by the last medical literature, did not recommend complementary radiation therapy.She reported at the Head and Neck Department in February 2019, due to otalgia and otorrhea from the left ear and complete facial paralysis; the physical examination showed a swelling at the site of the previous surgery.A CT of the neck with intravenous contrast, showed many colliquative areas in the parotid area, with lytic tissue extended to the external auditory canal, the tympanic cavity, and to the mastoid, with infiltration of the sigmoid sinus and erosion of the labyrinthine block up to the internal auditory canal and to the pontocerebellar angle Figure .A FNAB was performed and citology revealed a recurrence of AciCC of the parotid gland.The patient underwent a lateral skull base resection. A lateral approach to the skull base by pre‐sigmoid‐retrolabirinthine has been performed. The posterior and lateral semicircular canals have been skeletonized. The facial nerve was exposed in its second genu and the rest of the nerve was encased by the tumor; therefore, it was not possible to preserve it. The jugular bulb, very thin, started bleeding during the delicate dissection of the tumor, and bleeding was stopped by using Gelfoam. By introducing the endoscope, the clivus and the abducens nerve were on the view. A total excision of the tumor up to the left pontocerebellar angleThe histologic examination was in favor of a recurrence of AciCC Figure  , a new entity among salivary glands malignancy introduced in the 4th edition of the WHO classification of head and neck tumors.There are no clear characteristics of AciCCs found on CT, MRI, and ultrasound imaging.Management of AciCC, especially when metastatic, is very challenging: because of the rarity, data on management are based only on personal clinical experience and retrospective series. The preferred treatment of local disease is complete surgical excision, consisting in total parotidectomy.The role of radiotherapy remains controversial: the precise indications and oncologic effects of adjuvant radiotherapy in AciCC of the parotid gland are not well known, particularly in patients with negative, but close (<1 mm) margins without other high‐risk histopathologic factors.According to the most recent literature, the main factors that may predict poor outcomes are (1) extracapsular extension, (2) facial nerve involvement, (3) positive surgical margins, (4) cellular atypia, (5) high pathologic grade as evidenced by >2 mitoses/10 high power fields, (6) necrosis, (7) perineural and vascular invasion, (8) desmoplastic stromal reaction. To our knowledge, only 12 cases of AciCC of the parotid gland with lateral skull base involvement have been reported in literature.Breen JT et al.Buiret G et al.A study by Douglas JG et al.The patients described had the following characteristics  Table ; men to The mean time to develop lateral skull base disease was 15.8 years  (of two patients are not reported these data). This recurrence was treated by surgery alone in two cases (18.2%), with radiotherapy alone in three cases (27.3%), and with revision surgery followed by radiation therapy in three cases (27.3%); for three patients (27.3%) this item is not specified. No patient was treated with chemotherapy regime, confirming the lack of utility of chemotherapy against this histological type of parotid cancer.Four patients (36.4%) had distant metastases, while other four (36.4%) had cervical metastases. Most frequent skull base localizations were: stylomastoid foramen , petrous temporal bone , clivus , foramen ovale , cavernous sinus , sphenoid sinus .4Knowledge about AciCC has changed over the past few decades. Surgery is the first‐line treatment, while radiotherapy can be helpful in selected patients  and in case of recurrence. No chemotherapy regimen is currently being validated.From the literature, it emerges that a small number of patients will develop lateral skull base involvement, often years after the treatment of the primary disease. These subjects with cranial base disease are at a high risk for multiple late recurrences and even death.Although in literature there are no clear evidences about neurotrophicity of AciCC of the parotid gland, in our opinion, very rarely these tumors can grow up through the cranial nerves sheath developing within the posterior cranial fossa.Pietro De Luca: Conceptualization; data curation; formal analysis; funding acquisition; investigation; methodology; resources; validation; visualization; writing – original draft. Matteo Calvanese: Data curation; formal analysis; writing – original draft. Angelo Camaioni: Formal analysis; supervision; validation; writing – original draft. Giorgio Iaconetta: Formal analysis; supervision; validation; writing – original draft. Maurizio Iemma: Conceptualization; data curation; formal analysis; supervision; validation; writing – original draft.The authors have no conflict of interest to disclose. No fundings to declare.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Acute abdominal pain in adolescents has a multitude of diagnoses to consider ranging from life-threating ones to other less obvious. In this case report, a 15-year-old girl presented with right lower quadrant abdominal pain and tenderness one month after successful surgical management of acute appendicitis. Post-appendectomy abdominal pain could easily be attributed to post-operative complications, while, in reality, a different disease state may be the cause of the pain. Physicians should have a high index of clinical suspicion, even though the temporal association of events may suggest otherwise. Hemorrhagic ovarian cyst (HOC) should be included in the differential, as it was confirmed with imaging in our case. A conservative treatment approach with progesterone was chosen, with menses resuming 2 days later, leading to regression of the cyst. The clinical significance of this case relies on the timely recognition of a disease entity, in order to distinguish it from complications arising postoperatively. Acute abdominal pain is a common complaint in childhood and it poses a diagnostic uncertainty for clinicians, owing to the diversity of underlying surgical and non-surgical conditions. Acute appendicitis usually has a classic presentation, and hopefully, atypical manifestations are easily suspected, due to the high incidence of this surgical entity. However, the differential diagnosis of abdominal pain occurring after appendectomy is a major challenge for physicians, who should determine even uncommon conditions, apart from the most obvious ones, such as ileus and abscess . In thisPatient information: an otherwise healthy 15-year-old female visited the Emergency Department (ED) complaining of abdominal pain of two days duration localized in the right lower quadrant with progressing intensity. The pain was colicky in character, did not radiate, and was accompanied by a low fever of up to 37.5°C, nausea, and one episode of vomiting. The patient´s past medical and family history was unremarkable. She did not take any medications on a regular basis nor used tobacco, alcohol, or illicit drugs and had normal menstrual cycles since the age of 12 years old. Upon arrival at the ED, vital signs were normal. On physical examination, the abdomen was tender to palpation, especially in the right lower abdomen and the typical McBurney point was positive. The rest of the physical examination was unremarkable. Laboratory blood tests revealed leukocytosis with a left shift and increased C-reactive protein (CRP). Abdominal ultrasonography was performed that revealed a uterus and ovaries without pathologic findings and a small fluid collection within the lesser pelvic cavity. Right iliac fossa findings included an aperistaltic, non-compressible, dilated (>6 mm in diameter) and thick-walled mass, which was attributed to an edematous and inflamed appendix accompanied by hyperechoic peri-appendiceal fat and reactive iliac lymph node enlargement. The patient was admitted with the diagnosis of acute appendicitis, had intravenous fluid resuscitation, was started on antibiotics, and was taken to the theater for laparoscopic appendectomy. Intraoperative findings were unexpected. A pericaecal inflammatory mass  that was firmly attached to the lesser pelvic cavity was identified. Laparoscopic mobilization of the bowel loops, in order to reveal the appendix, was not possible and was considered unsafe, so the operation was converted. An open approach was performed, bowel loops were released and the appendix was found to be gangrenous in consistency with a rupture due to the presence of a fecalith. Visualization of the adnexa was normal. Following appendectomy, purulent material was aspirated, the peritoneal cavity was washed with normal saline and the operation was completed. The post-operative period was uneventful and the patient was subsequently discharged from the hospital with no perioperative complications. One month after the operation, the patient returned to the hospital due to new-onset abdominal pain and multiple episodes of vomiting of one-day duration.Clinical findings: vital signs were within normal limits and physical examination revealed moderate tenderness in the right iliac fossa without signs of peritoneal involvement.Timeline of the current episode: October 2021: admission to the hospital for appendicitis and subsequent appendectomy. After 10 of hospitalization, discharge with follow-up instructions. November 2021: re-admission due to hemorrhagic ovarian cyst (HOC), conservative treatment, and discharge with follow-up instructions.Diagnostic assessment: laboratory test results were unremarkable. Due to the patient´s recent appendectomy, imaging was performed to rule out complications associated with the surgery, like intra-abdominal fluid collection or ileus. Ultrasonography (nography  and compnography .Diagnosis: the results of imaging examinations were compatible with HOC due to the presence of cystic mass with internal septations and hemorrhagic elements.Therapeutic interventions: a more thorough patient history revealed that the patient had a 10-day delay in her usual menstrual cycle. Since her symptoms regressed and were not indicative of inflammation or acute surgical condition, and after obstetrician consultation, the mass was attributed to an ovarian hemorrhagic cyst and she was started on progesterone according to the patient´s age and weight. Two days later, she started menstruation, ultrasonography identified regression of the HOC, and the patient was discharged.Follow-up and outcome of interventions: ultrasound of the lower abdomen on the fifth day of menstruation, which was unremarkable.Patient perspective: when the patient was re-admitted she was concerned that she was coming across a possible post-operative complication. After the diagnostic work-up was completed and treatment was offered, she was fully relieved that her symptoms were due to menstruation abnormality.Informed consent: it was sought and obtained from the patient. Anonymity was maintained for confidentiality.Acute appendicitis is one of the most common surgical diseases in children with acute onset of abdominal pain. Pediatric surgeons should be familiar with recognizing not only the most typical symptoms but also evaluating accordingly the clinical presentation of a child, combined with a detailed history and the radiological findings, in order to avoid delayed diagnosis. In our case, the medical history and physical examination were suggestive of perforated appendicitis since the patient had been suffering from progressive and severe abdominal pain for 2 days prior to the ED visit, which was accompanied by abdominal rigidity .Given the recent operation at the right iliac fossa, the reappearance of the patient with another episode of acute abdominal pain could mistakenly be attributed to a postoperative complication, such as an abscess, ileus, or fluid collection, and easily lead to misdiagnosis. Post-operative complications of appendectomy are typically infectious, with the most common being wound infection and intra-abdominal abscesses, and are more common in perforated appendicitis, like in our patient ,5. To evHemorrhagic ovarian cyst is a common cause of intraperitoneal hemorrhage in women of reproductive age, but it is uncommon in early adolescence. The presenting symptom in most cases is lower abdominal pain, owing to irritating qualities of the hemoperitoneum, which commonly has a sudden onset, among other symptoms, like nausea or vomiting . A pregnHere, we presented a case of HOC, occurring shortly after appendectomy in a 15-year-old female. The typical clinical presentation of HOC may be difficult to differentiate from other causes, especially for pediatric surgeons, who are unfamiliar with this common gynecologic disease. Including HOC in the differential diagnosis of acute lower abdominal pain in young females is crucial, since its associated complications can be life-threatening. In the post-operative setting the clinician should always be in a position to first exclude possible post-operative complications but should always be vigilant of a new disease entity, such as HOC, that could be producing the new onset of complaints.


Xanthogranulomas are considered rare tumors, with their sellar and non-sellar frequency ranging from 1.6 to 7% among intracranial lesions, and described as a separate entity by the World Health Organization in 2000. The diagnosis of sellar xanthogranulomas is challenging, given their uncertain origin and clinical course. In addition, the limited reporting of sellar xanthogranuloma cases and the absence of characteristic images make these entities difficult to distinguish from other cystic lesions of the sellar region, such as adamantinomatous craniopharyngiomas, Rathke’s cleft cysts, pituitary tumors, arachnoid cysts, epidermoid cysts, and dermoid cysts. Here, we describe the clinical presentation, radiological findings, immunohistochemical/histopathological analysis, and the ultrastructural examination by transmission electron microscopy of five sellar xanthogranulomas cases reported in two care centers in Cordoba, Argentina. Two males and three females between 37 and 73 years of age (average 51.8 years) presented with persistent headaches, generalized endocrine defects, and visual problems. MRI revealed cystic formations in the sellar region, which usually projected into adjacent tissues such as the suprasellar region or cavernous sinuses, and compressed other structures such as the optic chiasm, pituitary gland, and cranial nerves. All patients underwent surgical intervention to remove the tumor tissue. The histopathological analysis of the samples showed cellular tissue with a xanthogranulomatous appearance, inflammatory cellular infiltrate (mainly lymphocytes and macrophages), fibroblasts, abundant collagen fibers, and hemorrhages. An ultrastructural analysis helped to identify cellular infiltrates and granules resulting from tumor cell activity. The data support the hypothesis that sellar xanthogranulomas could occur as an inflammatory reaction secondary to the rupture and hemorrhage of a previous cystic process, thereby generating an expansion of the tumor body toward adjacent tissues. The information obtained from these cases contributes to the current knowledge about this disease’s origin and clinical and histological evolution. However, the scarcity of patients and the observed phenotypic heterogeneity make its diagnosis still challenging. Undoubtedly, more investigations are needed to provide additional information in order to be able to achieve a more accurate diagnosis and effective treatment of this rare disease. Sellar xanthogranulomas (SX) are rare chronic inflammatory lesions secondary to hemorrhages, inflammation, infarction, and/or necrosis of a pre-existing Rathke’s cleft cyst, craniopharyngioma, or pituitary tumor . The incHistopathological findings usually reveal cholesterol spikes, hemosiderin deposits, chronic inflammatory cell infiltrates, multinucleated giant cells, macrophages, and fibrous proliferation . In addiTheir clinical presentation is frequently associated with headaches (50% of cases), visual defects (63–67%), central diabetes insipidus (33–43%), and anterior pituitary dysfunction 67%; . MoreoveThe preferable treatment for xanthogranulomas is the surgical reduction and decompression of the optic chiasm with preservation of the pituitary stalk . HoweverA 44-year-old female patient with a 1-year history of neck pain accompanied by headache. Cervical spine studies incidentally showed a tumor in the sellar-suprasellar region. A pituitary MRI revealed an expansive heterogeneous cystic, solid tumor lesion of 17 mm × 14 mm with regular contours. The cystic portion of the lesion showed high signal intensity on both T1 and T2 sequences. Additionally, a small eccentric left paramedian solid mural nodule of 8 mm × 5 mm is observed, which appears hypointense on T2. The tumor extends suprasellar and compresses the pituitary stalk and optic chiasm . The previa the transseptosphenoidal approach with an endonasal approach. The histopathological analysis showed an extensive xanthogranulomatous chronic inflammatory reaction composed of abundant clumped foamy macrophages . The thyroid function and androgen levels were normal. Cabergoline, 0.5 mg per week, was indicated. A pituitary MRI revealed a heterogeneous lesion at the adenohypophysis level measuring 12 mm × 17 × 20 mm, which was slightly hyperintense and with hypointense margins on T2 and isointense signal on T1, without any enhancement after intravenous contrast . The lesrophages , some murophages . At the rophages ,L. The prophages .sellae turcica and extended to the suprasellar cistern, compressing and displacing the optic chiasm. It also caused lysis and remodeling of the sphenoid bone in anterior and posterior directions while compressing both cavernous sinuses in a lateral direction . To date, the patient remains clinically asymptomatic, with no evidence of a deficit in the visual field and preserved pituitary axes, and is under expectant management.A 63-year-old female patient consulted due to intense persistent headaches for 15 days, located retro-orbitally, which did not respond to analgesics, and only partially responded to amitriptyline. The patient did not manifest any other relevant symptoms. Her personal history included arterial hypertension and dyslipidemia. The clinical laboratory did not show any alterations in the pituitary hormonal axes, and the computerized visual field was defect-free. A brain MRI exposed a spontaneously hyperintense image in the sellar region with a suprasellar extension, which suggested a craniopharyngioma or Rathke’s cleft cyst. The patient returned to control with the endocrinologist 6 years later with persisting chronic, left-sided, and retro-orbital headaches. In addition, she had noted a decrease in her peripheral vision in the months before the consultation. A pituitary MRI showed a voluminous lesion of 28 mm × 19 mm × 19 mm, hypointense on T1 and hyperintense on T2, with a thick-walled cystic appearance that occupied the entire irection ,B. The pirection ,D. In adirection . Nine moA 42-year-old male patient attended consultation due to 2 months of a persistent left temporoparietal headache without any visual alterations or relevant clinical history. A sellar MRI revealed an expansive intrasellar lesion with suprasellar growth toward both cavernous sinuses of 39 mm × 31 mm × 32 mm, which compressed the optic chiasm and the prechiasmatic portion of both optic nerves. It appeared as a solid lesion with an eccentric area, having a cystic appearance in the right lateral sector, and a hypointense signal on T1. The lesion included the left internal carotid in its cavernous course, the third, fourth, and sixth cranial nerves, and the V1-V2 segments of the trigeminal ,B. The cA 73-year-old male patient attended consultation with a history of colon cancer, ischemic heart disease, and bitemporal hemianopsia, with the latter being confirmed by the ophthalmology service after an episode of dizziness. A sellar MRI showed a solid, polylobed, space-occupying formation of 35 mm × 27 mm × 38 mm, which was visualized as isointense on the T1 sequence, heterogeneous on the T2 sequence, and predominantly hyperintense with some cystic areas. The lesion presented some hyperintense foci, which may correspond to hemosiderin or calcifications. It extended cephalad, obliterating the suprasellar cistern, generating a mass effect on the hypothalamus and the optic chiasm, displacing both optic nerves, invading both cavernous sinuses, and surrounding the internal carotid arteries ,B. A horActive tumoral cells, with numerous mitochondria and small secretory granules aligned to the plasma membrane, and tumoral necrotic cells next to connective tissue, were also detected .The SX are sporadic intracranial lesions and only a few cases have been reported in the literature . In thisOur case series was characterized by presenting a wide range of xanthomatous lesions, either isolated or associated with pituitary adenomas. The average age of presentation was 51 years, consistent with previous reports , and thrIn the present study, we have described the radiological findings, which were typical. However, imaging features may overlap with other sellar and suprasellar neoplasms, making it challenging to establish a definitive diagnosis based on imaging alone. In our case series and, in agreement with reports from Macroscopical features of SX include a fibrous capsule densely adherent to the dura, and a tumoral mass with non-hemorrhagic soft tissue and cystic components . This maIn the initial description of SX, The contribution of electron microscopy is an aspect worth highlighting from this present study, which provided valuable information from ultrastructural details and exposed some of the etiological factors that trigger the aforementioned inflammatory process. This approach allowed the precise identification of ultrastructural features in both the proliferating epithelial compartment and immune infiltrating cells . After nThe pathogenesis of SX is controversial. The histopathological characteristics and the evident presence of chronic inflammatory cells have fueled the theory that SX are on the same spectrum as Rathke’s cleft cysts and craniopharyngiomas, representing a potential side reaction caused by repeated swelling and bleeding. In fact, it often originates with the rupture of a cyst, such as Rathke’s cleft cysts, leading to the recruitment of inflammatory cells and macrophage activation. This is accompanied by local tissue damage, lipid release, and cholesterol crystal formation, thus perpetuating the granulomatous response. Craniopharyngioma, usually the adamantinomatous subtype, is one of the neoplasms that can have degenerative changes, such as cystification, calcification, and xanthogranulomatous reaction . SimilarOn the other hand, SX are not specific to the sellar region, which motivated previous classifications to include them into the category of “xanthomatous reactions of the nervous system,” alongside other conditions of metabolic, degenerative, and reactive origins, which are usually non-neoplastic pathologies with similar morphological characteristics .To summarize, it is still debated whether xanthogranuloma is an entity or a culminating, reactive, secondary inflammatory process in another as yet unidentified lesion . TherefoFinally, the prognosis of sellar xanthogranuloma is usually favorable. Since the long-term behavior of xanthogranulomatous pituitary lesions after surgical removal has not been evaluated, we recommend conducting close clinical and radiological follow-up, as has also been suggested by By closely monitoring patients postoperatively, we can gain a better understanding of the natural history of sellar xanthogranuloma and establish appropriate non-surgical therapeutic goals.The frequent association of xanthogranulomas with other tumor lesions in the sellar region, whether or not neoplastic, suggests a reason for variations observed in the clinical-imaging spectrum of SX. At the same time, this demonstrates the histological heterogeneity of this clinical entity, presenting isolated xanthogranulomatous lesions and others associated with well-characterized tumors.To conclude, our work provides new and valuable clinical, radiological, histopathological as well as ultrastructural information from five new cases reported in central Argentina. Added to the few previously reported cases, they contribute to obtaining a better understanding of the symptoms, pathogenesis, and development of SX, in order to achieve a more efficient and effective diagnosis and therapeutic action.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving humans were approved by the Local Ethics Committees of the corresponding medical centers: CIEIS—Clínica Universitaria “Reina Fabiola” and Sanatorio Allende of Cordoba, Argentina. All patients signed a written informed consent before surgery. This project was registered at the National Registry of Health Research (RePIS No. 3974). The studies were conducted in accordance with the local legislation and institutional requirements. The participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.SCF and MCB contributed to the conception and design of the project, sample collection, and manuscript writing. ACV and EG acquired and analyzed the imaging data. MLB, MJP, EM, RAD, CSS and EJH collected the clinical and MRI data. CAM and AAQ acquired the electronic microscopy images and described the immune cell ultrastructures. ALDP wrote and approved the final manuscript. CAM, AAQ, FNP, and ALDP reviewed the manuscript and contributed to quality control. All authors contributed to the article and approved the submitted version.This work was supported by the following funds granted to ALDP: Consejo Nacional de Investigaciones Científicas y Técnicas , Secretaría de Ciencia y Tecnología, Universidad Nacional de Córdoba , and Agencia Nacional de Promoción Científica y Tecnológica—Ministerio de Ciencia y Tecnología (FONCYT-PICT 0950-2020-2024).The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Point-of-care ultrasound (POCUS) is becoming more prevalent in recent years for evaluating patients presenting with musculoskeletal injuries in the emergency department (ED). This imaging modality has been utilized to diagnose soft tissue and bony injuries accurately, obtain appropriate consultation, and perform timely interventional procedures in the ED.We present the case of a 55-year-old man who presented to the ED with significant left ankle pain following a ground-level fall. His physical examination showed swelling and tenderness around the ankle. POCUS examination aided the rapid and accurate detection of acute Achilles tendon rupture.This case demonstrates that POCUS is a valuable diagnostic tool in evaluating patients with a suspected Achilles tendon rupture, especially in a resource-limited setting. Point-of-care ultrasound (POCUS) is becoming more prevalent in recent years for evaluating patients presenting with musculoskeletal injuries in the emergency department (ED). POCUS has been utilized to diagnose soft tissue and bony injuries accurately, obtain appropriate consultation, and perform timely interventional procedures in ED , 2. ReceA 55-year-old man with a history of diabetes mellitus and seronegative rheumatoid arthritis presented to the ED of the Aga Khan University Hospital (AKUH), Karachi, with a complaint of pain and swelling in the left ankle and inability to bear weight on his left lower limb after a history of ground-level fall 2 h back. He initially went to an outside hospital, where an X-ray of his foot showed an avulsion fracture of the calcaneal bone Fig. .Fig. 1X-He was referred to AKUH for an orthopedic opinion. On arrival, he was vitally stable. The general physical and systemic examination was unremarkable. His musculoskeletal examination showed significant swelling and tenderness around the left ankle and difficulty bending the foot downward (plantarflexion) Fig. . He had The on-call ED consultant performed a POCUS. The patient was lying prone with his feet hanging off the bed. A high-frequency linear probe was selected and placed on the posterior ankle in the long axis in a neutral position, which showed the disruption of the typical linear tendon fibers. Dynamic testing revealed the movement of the distal stump of the Achilles tendon adjacent to hypoechoic fluid, most likely reactive edema or blood from the acute rupture. Hyperechogenicity was also noted at the proximal end of the Achilles tendon, with acoustic shadowing probably representing an avulsed fragment of the calcaneum Fig. .Fig. 3PoHis foot was splinted in the plantar flexion position, and an orthopedic follow-up was arranged in two days. His MRI (magnetic resonance imaging) of the foot was done on an outpatient basis as advised by the orthopedic consultant, who was reported as an “avulsion fracture of the calcaneum at the attachment site of the Achilles tendon with retraction of the partially ruptured Achilles tendon. The retracted ruptured end of the Achilles tendon is at the level of the distal articular margin of the tibia and is approximately 47mm from its attachment site. Significant surrounding soft tissue edema and hematoma are noted.” He was treated conservatively with analgesics and non-weight-bearing status.An Achilles tendon rupture diagnosis in the ED is often made solely based on history and physical exam; however, up to 20% of ruptures go unnoticed during the hyperacute phase and are frequently misdiagnosed as an ankle sprain . ClinicaPOCUS is a useful ancillary tool for evaluating soft tissue injuries in real time and at a lower cost. The sensitivity of ultrasound for detecting this type of injury ranges from 79 to 100%. The clinical utility of ultrasound in Achilles tendon injuries has previously been demonstrated –14. It eOur case demonstrates the importance of POCUS in early decision-making and management of patients in a resource-limited country. Literature has shown that the POCUS has a positive impact on clinical decision-making in resource-limited settings, resulting in a decreased length of stay in EDs and a measurable reduction in planned referrals , 17. MorPOCUS is a practical and valuable modality to diagnose Achilles tendon rupture in patients presenting to the ED with a history of an ankle injury at the bedside. In combination with clinical history and examination, this can reduce ED stay and the number of expensive and time-consuming imaging resulting in cost-effectiveness, early decision-making, and planned referrals. POCUS is especially well-suited for resource-limited settings due to its quick accessibility of results and independence from additional infrastructure and staff (radiologists). As advancements continue, affordability and portability will further ease the adoption of POCUS over time.


Angiokeratoma is a rare vascular cutaneous disorder that usually presents as mostly asymptomatic aside from multiple dark red to blue or black papules over the skin in several clinically distinct conditions. Very rarely, it occurs in solitary localized forms that clinically mimic vascular disorders or sometimes melanoma. Solitary cutaneous angiokeratoma may result from damage to a venule's wall in the papillary dermis. This case study describes a 28-year-old male with a single angiokeratoma on the lateral aspect of his upper thigh and a clinical suspicion of a cutaneous melanocytic tumor. This case is intended to raise awareness about such rare skin lesions and the importance of histopathological examination. Angiokeratoma (AK) is a benign tumor composed of dermal ectatic capillaries with overlying acanthotic and hyperkeratotic epidermal tissue. It can appear as a variety of vascular keratotic black lesions that differ in size, depth, and site . There aClinically, solitary AKs sometimes mimic angiomas or hematomas and show no distinguishing clinical features in comparison with these frequent skin lesions. However, some cases can raise suspicion for melanoma or dysplastic nevus, especially when the vessels are thrombosed. Thus, the way to reach an accurate diagnosis is a histopathological analysis based on the classical changes occurring in the epidermis overlying the ecstatic vascular channels, this ectasia in the papillary dermis is considered the main pathogenesis of this disorder ,5. We reA 28-year-old male presented with a reddish-to-blackish elevated lesion that started on the upper lateral part of the right thigh and had been increasing in size for a year. The lesion was slowly progressing, lately painful, and reached more than 1 cm in size with an erythematous halo. The patient had no history of similar lesions and no history of trauma or injury to the affected site.Clinical examination revealed a solitary well-defined red-black plaque with a maximal diameter of 12 mm and no tenderness or ulceration. The lesion was firm in consistency and showed a halo with erythematous skin on the periphery  underlying hyperkeratosis. With the exception of AK circumscriptum, which results from malformation of dermal capillaries, AKs are a consequence of ectatic dilation of pre-existing capillaries in the papillary dermis . The firAKs of the scrotum and vulva (AK of Fordyce) are single or numerous lesions that range in color from red-purple to black and develop along superficial vessels of the scrotum and vulva. Scrotal lesions may also be related to inguinal hernias, varicoceles, and thrombophlebitis. Oral contraceptive use, vulvar varicosities, hemorrhoids, and elevated venous pressure during pregnancy can all be linked to vulvar lesions. AK corporis diffusum is characterized by a few to numerous, frequently grouped AKs that usually affect the lower trunk area . They are known to occur in healthy people as well as those with various enzyme-deficiency illnesses, such as Fabry disease. AK of Mibelli is composed of multiple hyperkeratotic papules and plaque, which are most frequently found on the lateral and dorsal sides of the fingers and toes. They can also affect the hands and feet. Chilblains and acrocyanosis could be associated with AK of Mibelli. AK circumscriptum is characterized by numerous distinct papules or hyperkeratotic papules and nodules that are frequently confluent. They are most often unilateral and affect the trunk, arms, or legs. Lastly, solitary or multiple AKs are single or multiple well-defined plaques that are red-black in color and commonly found on the lower limbs. Solitary lesions can be mistaken for melanoma -11. AlthHistological diagnosis is an important step in diagnosing and detecting unusual skin lesions all over the body, including the upper thigh and perianal area, which may be neglected or missed ,14. HistSolitary AKs and targetoid hemosiderotic hemangiomas (THHs) are acquired vascular malformations caused by superficial vascular dilatation that may result from trauma. On the lower limbs, they both frequently develop a brownish or blackish papule that looks like a melanocytic lesion. Both have extravasated red blood cells, hemosiderin deposits, ectatic papillary dermal vessels, lymphocytic infiltrates, and lymphangiectasias with overlaying hyperplasia. THHs are larger, have more pronounced dermal vessel alterations, often have dissected vascular empty spaces, and have greater hemosiderin deposits than solitary AKs .Surgical excision is the most common and effective treatment for AKs. The two goals of this procedure are simultaneously removing the tumor and confirming the diagnosis . Other tWe have presented an uncommon case of a solitary cutaneous AK in the upper lateral part of the thigh. The lesion was clinically suspected to be a melanocytic tumor but was histologically verified to be AK without any other accompaniment pathological lesions. The lesion was effectively excised under local anesthesia, and there was no clinical recurrence. Histopathology examination is crucial for any excised skin lesion.


Echinococcus. The liver is the most common location for a primary echinococcosis. However, the parasite may bypass or spread from the liver to the lungs, causing primary or secondary pulmonary echinococcosis, respectively. Pulmonary echinococcosis is a clinically challenging condition in which anthelminthic regiments are important, but surgery has the central role in removing the cysts and preventing recurrences. Surgical treatment may involve cystotomy, enucleation, capitonnage, or atypical resections, which occasionally are in combination with hepatic procedures. The utilization of modern devices is greatly underdescribed in surgery for thoracic infections, even though these facilitate much of the work. Therefore, this article aims to describe pulmonary echinococcosis and the role of modern surgical devices in the treatment process. Furthermore, we report surgical treatment of three different cases of pulmonary echinococcosis. Surgeries of uncomplicated and ruptured hepatic or pulmonary cysts are described. Simple small pulmonary echinococcal lesions can be excised by endostaplers both for diagnostic and curative reasons. Larger cysts can be removed by energy devices unless large bronchial air leaks occur. Complicated cysts require treatment by more extensive techniques. Inexperienced surgeons should not abstain but should carefully decide preoperatively how to proceed.Human echinococcosis is a zoonotic infection caused by the larvae of the tapeworm species  Echinococcus (E.). Two of the four known pathogenic species to humans, canine (E. granulosus) and fox tapeworm (E. multilocularis), cause cystic echinococcosis (CE) and alveolar echinococcosis (AE), respectively [Human echinococcosis is a zoonotic infection caused by the larvae of the tapeworm species ectively . Humans ectively . Primaryectively . The aimA patient with multiple bilateral lung lesions was diagnosed after investigations for a large hepatic lesion that turned out to be echinococcosis. A 41-year-old male patient reported abdominal pain and shortness of breath and was referred to the hospital by a primary care physician. Primary radiological investigations revealed a large hepatic lesion and multiple bilateral pulmonary lesions. Magnetic resonance imaging (MRI) was performed to evaluate the hepatic lesion , while cThese were addressed synchronously with bilateral posterior thoracotomies in a prone position, using our experience in bilateral lung transplantation with this approach . Gentle manual palpation is necessary for localization. Wedge-type excisions were performed using standard automatic endostaplers of 45/60 mm length on clear macroscopic margins of minimum 1 cm interval . HistopaIn the presented case, the hepatic lesion ruptured directly to the right lower segmental bronchus S10 as confirmed acutely in a flexible bronchofibersocopy, where large amounts of purulent discharge were lavaged. Chest CT showed communication of the ruptured lesion with the large right airways . This acThe uncomplicated large intraparenchymal pulmonary lesion. Typical diameters can exceed 15 cm. In such cases, wedge-excisions are unsuitable due to the size and a potentially central location. Instead, enucleation is indicated to spare parenchyma. The dissection of the cyst from the surrounding parenchyma is facilitated with a standard thoracoscopic energy device with ‘coagulate and cut’ functions and dissecting maneuvers, similar to adhesion lysis . CarefulThe complicated large cyst. A large cyst that may rupture during dissection either when using a dissector or an energy device. In this unfortunate case, raw parenchymal surfaces may bleed, produce large air leaks, or leave redundant tissue. In this case, endostaplers or energy devices do not offer proven advantages. Instead, suturing  should eliminate the space after careful debridement .E. granulosus is considered endemic in pastoral regions, such as Argentina, Chile, Central Asia, Africa, and Mediterranean countries. Single-organ involvement is a more frequent manifestation with the liver being the most common location (approximately 70%). Lung involvement is observed in 20% to 30% of CE patients [Infection of patients . Neverthpatients . The parpatients . The majpatients . In contpatients . Furtherpatients . Clinicapatients .E. multilocularis, is an endemic infection in North America, Central Europe, and East Asia. The parasite forms primary cysts almost exclusively in the liver. The infection with E. multilocularis resembles a malignant disease. It may be asymptomatic for up to 15 years of incubation. However, rapid growth of AE lesions has been identified in immunosuppressed patients, as demonstrated in reports of infected patients after solid organ transplantations [E. multilocularis may give metastasis to other organs [On the contrary to the CE, alveolar echinococcosis, caused by the ntations ,12. Subsr organs . Accordir organs . Therefor organs . AE is ar organs . Howeverr organs .In pulmonary pathologies, X-ray is usually a primary method of investigation. Since pulmonary echinococcosis may be asymptomatic for a long time, the parasitic cysts are often diagnosed incidentally. CT is usually used to evaluate abnormal lesions detected in chest X-ray. According to a study by Wu et al., pulmonary CE lesions are usually single (60.9%) with an average size of 7.9 cm. The authors suggest that the loose pulmonary tissue and rich circulation may contribute to the fast growth of the cysts . On the Despite clinical course and radiological imaging, serology, histopathology, and molecular investigations may help in differentiating between CE and AE. Serological investigations often involve the detection of specific IgG antibodies . FurtherEchinococcus are required. Several studies suggest that a variety of long known drugs might inhibit the growth of the parasite. These agents include mefloquine, pyronaridine, atovaquone, 5-fluorouracil, and metformin, among others [Currently, there are three therapeutic strategies for hepatic CE: surgery, ultrasound-guided aspiration, and chemotherapy ,34. Watcg others . Interesg others .E. granulosus and E. multilocularis infections [In pulmonary lesions, surgery aims to remove the parasite, prevent intraoperative dissemination, and preserve pulmonary parenchyma. Kavukcu et al. reviewed 1118 surgeries for pulmonary hydatidosis. Posterolateral thoracotomy was performed in 98.3% of patients. On the other hand, only 3.3% of patients underwent two-stage bilateral thoracotomy. Furthermore, only 3.3% of patients developed hydatid recurrence. Empyema was diagnosed in 0.3% of patients and these patients were treated with antibiotics and drainage . Aydin efections ,48,49.Despite advances and measures for zoonosis prevention and treatment, PE remains a clinical reality. Thoracic surgeons in non-endemic echinococcosis areas lack the experience in established procedures suitable for managing PE such as cystotomy, enucleation, capitonnage, or atypical resections, which occasionally are in combination with hepatic procedures ,51. If eDue to non-specific symptoms and various manifestations in radiological imaging, there is a broad differential diagnosis for pulmonary echinococcosis. To begin with, tumors found on imaging in heavy smokers raise the suspicion of pulmonary neoplastic disease . FurtherE. multilocularis, while patients 2 and 3 with E. granulosus. Furthermore, histopathological examination of echinococcal lesion of the first patient revealed central necrosis, granulation tissue, and infiltration with neutrophils and lymphocytes, which points to alveolar variant of the infection. Patients with pulmonary echinococcosis may present symptoms such as cough, dyspnea, chest pain, haemoptysis, and fever, among others. Furthermore, radiological imaging may present solitary or multiple round lesions. As a result, the manifestation of pulmonary echinococcosis may resemble a primary or metastatic malignant disease, especially in patients with previous neoplasms or in heavy smokers. Furthermore, radiological examination may suggest the presence of other benign and rare pulmonary pathologies. The differential diagnosis of such lung tumors should include PE. Simple pulmonary echinococcal lesions up to 5 cm can be excised by endostaplers both for diagnostic and curative reasons. Larger cysts can be excised by energy devices unless large bronchial air leaks occur. Lobectomy may be required if a cyst is too large or located centrally. Complicated cysts require treatment by more extensive techniques. Inexperienced surgeons should not abstain but should carefully decide preoperatively how to proceed.Pulmonary echinococcosis is an uncommon but significant clinical entity. Differentiating between CE and AE is necessary for proper patient management and various methods may help in distinguishing between these two infections. In the presented cases, all the preoperative diagnostic workup has been completed by the referring internal medicine departments following the existing institutional guidelines valid at that time. Patients’ origin , clinical course, radiological imaging, and intraoperative images suggest that the first patient was infected with


Myofibroblastic tumors are extremely rare in the larynx, with just over 40 published cases. Despite being a benign tumor, it presents with a marked inflammatory character, local destruction, and the possibility of degeneration to malignant histological types with metastatic capacity. Anatomopathological differential diagnosis is fundamental in these cases. Myofibroblastic tumor is a benign tumor with a high capacity for local destruction, malignant degeneration, and the possibility of metastasis. Anatomopathological and immunohistochemical studies are essential for diagnosis. They may cause dysphonia, dyspnea, pharyngolaryngeal foreign body sensation, or dysphagia, among others.2We present the case of a 39‐year‐old man who had been seen at another center with clinical symptoms of dysphonia that had been evolving for months. He was referred for pathological findings of an IMT of the right vocal cord after a biopsy by direct laryngoscopy. The patient had no personal records of interest or toxic habits. He was non‐smoker and non‐drinker. He did not report any consumption of illicit substances. The blood count and coagulation were normal, with the only abnormal finding in the biochemical study: being LDL cholesterol 280 mg/dL. During the examination, the patient presented symptoms of dysphonia and pharyngolaryngeal foreign body sensation along months of evolution. The fibrolaryngoscopic examination revealed a lesion with the appearance of a pedunculated angiomatous polyp <1 cm in its longest axis, in the anterior third of the right vocal fold, without involvement of the anterior commissure  on chromosome 12, t, , tropomyosin 4 (TPM 4), TPM 3, t, cysteinyl tRNA synthetase, and Ran‐binding protein. In addition, TP53 positivity has been demonstrated in some cases, which could have prognostic influence, being more common in recurrent cases, with transformation to malignant histological types or production of metastases.Despite being very infrequent with its lesions in the larynx, IMT is a pathology that has to be considered, requiring similar procedures as laryngeal neoplasms, where biopsies need to be taken to establish a clearer anatomopathological diagnosis for defining a treatment plan. The first‐line treatment is surgery, either with cold instruments or resection with laser surgery. Radical excision with free edges is the critical prognostic factor. Radiochemotherapy is reserved for cases with positive margins or a formal contraindication to surgery. A close clinical follow‐up of this pathology is essential, with most series requiring it for at least 12 months, given the variable risk of recurrence and distant metastases.Given the marked inflammatory component of the lesion, a second‐line corticosteroid therapy is widely validated in cases with contraindications for surgery or tumor recurrence, and it is also used for head and neck IMT in locations other than the larynx.Genetic alterations are opening new lines of research: series of treatment with ALK inhibitor crizotinib have been published in cases of ALK‐positive ITM, as a second‐line therapy for those cases of contraindication to surgery, partial resection, or positive surgical margins.5IMT is an infrequent pathology in the larynx, included in the differential diagnosis of benign laryngeal pathology. Despite this, it is essential to consider the locally expansive and inflammatory nature of this lesion, the risk of transformation to malignant histological types, and the possibility of distant metastases. Anatomopathological and immunohistochemical studies are fundamental in the differential and definitive diagnosis. The first‐line treatment is surgical resection, completed with free margins, and close postoperative follow‐up given the possibility of local and distant recurrence.Bazan Inostroza B.: involved in writing and data collection, involved in the clinical and in the surgical procedure and follow‐up of the clinical case. Prada Jorge, Eisenberg Gustavo, and Raboso Eduardo: involved in supervision and review, involved in the clinical and in the surgical procedure and follow‐up of the clinical case. Adrados Magdalena: involved with the histology images and histological analysis of the surgical specimen.There are no conflicts of interest.The authors of the following article declare that there is no conflict of interest. No funding was required. The project has been approved by the ethics committee of the Hospital Universitario de La Princesa . The patient has signed the informed consent for the study to be carried out.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy. Written informed consent was provided by the patient for publication of this case and the images.