Pulmonary nodules often present a diagnostic challenge due to their diverse etiology, ranging from benign to malignant conditions. We discuss the diagnostic journey of a 71-year-old female patient with a history of kidney stones, who was incidentally found to have a pleural-based pulmonary nodule during a CT urogram. Subsequent imaging showed nodule growth, prompting further investigations, including a PET/CT scan and CT-guided biopsy, which yielded inconclusive results. A multidisciplinary approach recommended surgical resection, revealing three mobile calcified-like nodules within the pleural space, later identified as hyalinized nodules. The absence of malignancy was reassuring.These benign, mobile pleural bodies, known as thoracoliths, are challenging to differentiate from pulmonary nodules. This case underscores the importance of considering rare benign entities in pulmonary nodule differentials and highlights the need for a multidisciplinary approach, surgical intervention, and open-mindedness in complex diagnostic scenarios. Pulmonary nodules have long been considered a vexing challenge in clinical practice, embodying a complex diagnostic dilemma. Encountered frequently, these discrete lesions in the lung parenchyma can be harbingers of both benign and malignant conditions. Their evaluation necessitates a nuanced approach that aims to rule out malignancy while avoiding unnecessary invasive procedures. This case report delves into an instance of this diagnostic conundrum, involving a 71-year-old female patient with a history of kidney stones. Her initial presentation stemmed from the incidental discovery of a pulmonary nodule during a routine CT urogram in July 2022. The sequence of events over the next few months illustrates the critical need to consider rare benign entities, such as thoracoliths, within the differential diagnosis of pulmonary nodules that can closely mimic malignancy.Pulmonary nodules are a common radiological finding, often detected incidentally during imaging studies for unrelated health concerns, as was the case with our patient. The challenge lies in differentiating between these nodules, which can encompass a broad spectrum of etiologies, ranging from benign granulomas, infections, and inflammatory processes to potentially life-threatening malignancies like lung cancer. Timely and accurate diagnosis is crucial as it dictates the subsequent clinical management and interventions.The clinical significance of pulmonary nodules is underscored by the ever-evolving landscape of medical imaging technologies. High-resolution CT (HRCT) scans, PET/CT, and MRI have revolutionized the detection and characterization of these nodules, thereby enabling more precise assessment of size, shape, density, and metabolic activity. However, even with these advanced diagnostic tools at our disposal, the quest for an accurate diagnosis remains fraught with complexity as illustrated in our case discussed below.Our patient initially underwent a CT urogram in July 2022 to assess her kidney stones, which revealed an incidental pulmonary nodule, setting in motion a series of investigations. Subsequent CT chest scans at three and six months from her original CT urogram revealed a concerning trend: the right lower lobe nodule was growing in size, increasing from 12 to 16 mm (Figures A year later, a PET/CT scan was performed to evaluate the metabolic activity of the nodule Figure . It indiThe complexity of the case necessitated a multidisciplinary approach. In light of the increase in nodule size and the inconclusive biopsy findings, a tumor board recommended surgical resection as the most appropriate course of action. This decision led to a robotic surgical procedure using the Da Vinci Xi system in May 2023.Intraoperatively, three free-moving round nodules at the level of the right lower lobe in the pleural space Figure  were fouThe patient was followed up three months later and underwent a repeat CT chest, which showed no residual disease Figure ; she is Pulmonary nodules represent a diagnostic challenge due to the myriad possible underlying causes. They can range from benign lesions to malignant tumors, and distinguishing between them is paramount for appropriate clinical management. In this case, the pivotal discovery was not made until the surgical intervention. Intraoperatively, the surgical team identified three intriguing entities within the pleural space at the level of the right lower lobe: free-moving calcified-like nodules Figure . These nPathological analysis of the resected nodules revealed a well-defined structure characterized by densely eosinophilic collagenous material Figure . These nVarious theories have been proposed regarding the origin of these thoracoliths, including degenerated pleural lipomas  (a rare Our case is unique in that it involved right-sided thoracoliths mimicking pulmonary parenchymal nodules.This case report highlights the intricate and sometimes unpredictable nature of pulmonary nodules. The initial discovery of a nodule during a routine imaging study led to a series of investigations, each presenting unique challenges and raising concerns about malignancy. The ultimate identification of hyalinized nodules, or thoracoliths, within the pleural cavity served as a valuable lesson in considering rare benign entities when confronted with pulmonary nodules that may mimic lung cancer. The case also underscores the importance of a multidisciplinary approach, as well as the necessity for close collaboration between clinicians, radiologists, and pathologists when assessing pulmonary nodules. Furthermore, it lays stress on the critical role of surgical intervention in reaching a definitive diagnosis in complex cases where noninvasive methods prove inconclusive. As healthcare providers, we must remain vigilant and open to the possibility of unexpected findings, even when encountering radiological and clinical evidence that may suggest a more ominous diagnosis.


Lymphatic malformations are a rare pathology that presents a highly variable clinical expression. Intraorally, it mainly affects the dorsum of the tongue. The objective of this work is to present a case of lymphatic malformation in an atypical location. A 20-year-old male who attended the clinic for multiple vesicular lesion in attached gingiva, asymptomatic and of unknown evolution. Removal of the lesion and subsequent histological analysis were performed, which showed a microcystic lymphatic vascular lesion. Immunohistochemistry for D2-40 was performed, which corroborated the lymphatic origin of the lesion. At 6 months, no recurrence of the lesion was recognized. Clinicians should include lymphatic malformations in the differential diagnosis of multiple vesicular lesions. Knowing the oral manifestations of this entity is essential for its proper diagnosis and clinical management. Key words:Gingiva, oral lymphatic malformation, diagnosis. Lymphatic malformations (LM) or lymphangiomas are an uncommon pathology, characteristically affecting the body areas that are richest in lymphoid tissue, including the head and neck region, armpits and mediastinum ,2. SpeciLymphatic malformations are currently considered a congenital developmental disorder, rather than a neoplasm per se ,4. In faFrom a structural point of view, lymphatic malformations can be classified as microcystic, macrocystic or mixed ,5. At inClinical manifestations will depend on the type of lymphatic malformation (macro/microcystic), its depth and location. In the oral cavity, they usually appear as clusters of vesicular, raised, soft and depressible lesions, usually of the same colour as the mucosa or with a yellowish or even erythematous appearance, due to hematic extravasation. Although it is true that, in cases of deep lesions, they may present as a diffuse increase in volume .The diagnosis of this entity is eminently clinical, and histopathological study is necessary for its confirmation. Occasionally and especially in the case of deep and extensive lesions, imaging tests are required to assess the extent of the lesion, with magnetic resonance being the most indicated imaging technique. In cases of superficial lesions, the use of ultrasound techniques may be of interest.The differential diagnosis should mainly include other vascular pathologies, such as haemangiomas or blood vessel malformations. At intraoral level, benign epithelial lesions, such as squamous papillomas, may occasionally be included .The clinical management of lymphatic malformations will depend on the extension and location of the lesion, as well as the patient’s age. Two basic types of treatment have been described for the management of vascular malformations, which are surgical treatment and the use of different sclerosing agents .The aim of this article is to describe the clinical and histopathological characteristics of a diagnosed case of oral lymphatic malformation, as well as to describe its surgical management.20-year-old male, who attended the clinic for assessment of an asymptomatic lesion on the vestibular gingiva detected during dental examination. The patient did not recognise any traumatic factors associated with the appearance of the lesion. Since he became aware of the appearance of the lesion, he indicated no significant changes.The patient had no medical history of interest, nor did report any known allergies. He indicated that he was a non-smoker and occasional alcohol consumer.Clinical examination revealed a lesion showing multiple exophytic vesicles, located in the attached vestibular gingiva, at the level of the papilla between 4.4 and 4.5. The lesion was partially pedunculated presenting irregular surface and slightly erythematous colour Fig. . On palpWith all these data, lymphatic malformation was our presumptive clinical diagnosis, including localized juvenile spongiotic gingival hyperplasia in the differential diagnosis.Given the clinical characteristics of the lesion, it was decided to perform its removal and subsequent histopathological analysis. Under infiltrative anaesthesia using 4% articaine with 1:100.000 adrenaline and with a No. 3 scalpel and No. 15 blade, surgical exeresis was performed, leaving the surgical bed healing by secondary intention Fig. .Histopathologic study revealed a subepithelial vascular lesion consisting of multiple small vessels and thin endothelium Fig. A. These At the 7 days follow-up, adequate healing was observed, with no functional alterations Fig. . No recuThe case presented shows the clinical and histopathological expression of a microcystic lymphatic malformation in attached gingiva. Even though the gingiva is not the typical location of LM in the oral cavity, it has been reported to constitute about 5% of documented cases (Although the actual prevalence of lymphatic oral malformations is not known, the diagnosis of this condition is between 0.02 and 0.16% of the biopsies analysed in oral pathology laboratories .et al., published in 2022, describes the activation of PI3K/AKT and MAPK/ERK intracellular pathways (In recent years, several molecular alterations related to the appearance of lymphatic malformations, both sporadic and those involved in genetic syndromes, have been reported ,8. The mpathways .et al., indicated that only 30.3% of the cases of oral lymphatic malformation had a clinical diagnosis of this entity (Meirelles s entity . This fas entity . In the s entity -13, as ss entity -13, simiIn any case, therapeutic management is similar, albeit the histopathological characteristics are what would help differentiate both entities.LM can acquire very different sizes, although most intraoral lesions are smaller than 1cm , as in tIn relation to the clinical management of the case, in our case, due to the size and location, we decided to proceed with surgical removal and its subsequent histopathological study, which is the treatment of choice for small and superficial lesions. On the other hand, in larger lesions and where there may be a post-surgical functional problem, treatment with sclerosing agents may be considered, including bleomycin, OK-432, alcohol, polidocanol or others ,15.In conclusion, we may indicate that lymphatic malformations are a rare pathology of the oral mucosa that can be expressed by the presence of multiple vesicular elements of translucent or reddish colouring and should be included in the differential diagnosis of vascular lesions. It is important to know the characteristics of this oral pathology in order to establish a proper diagnosis and clinical management.


A 34‐year‐old woman with complaints of mandibular swelling that started 4 months earlier was referred to the Oral Diseases Department. Based on the clinical and radiographic appearance, the primary diagnosis was an intraosseous reactive lesion. However, the result of histopathology indicated osteosarcoma. The diagnosis of osteosarcoma is sometimes challenging due to similarity of its clinical manifestation to those of benign lesions. Correct diagnosis and timely referral have a major impact on prognosis and patient survival. The patient had no particular habits, no systemic disease or taking certain medications and his psychological status was normal. 4 months earlier, the patient was referred to a general dentist with pain in the lower right area and had a pulpotomy of the lower right fourth tooth. The patient also noted a brief loosening of the same tooth 20 days later and an exophytic lesion in the vestibule of the area. On the second referral to a general dentist and with a possible diagnosis of dental infection, the patient was treated with the antibiotics cephalexin and metronidazole for 2 weeks with no improvement. According to the patient, the size of the lesion almost tripled in 4 months. On extraoral examination by an oral and maxillofacial specialist, facial asymmetry was observed in the form of a brief swelling with a firm consistency in the left cheek toward the lower border of the mandible. No lymph node involvement was observed on examination of the neck.On intraoral examination, there was an exophytic dome‐shaped lesion with an approximate size of 2.5 × 3 cm in the buccal vestibule from the distal lower right second tooth to the distal lower right fifth tooth. The color of the mucosa covering the lesion was normal with some telangiectasia. The lesion was uniformly firm with bone thinning noticeable upon touch in some areas. The lower right third tooth showed a grade 2 loosening and the lower right fourth and fifth teeth showed a grade 1 loosening. The patient also had pain upon touch and dental occlusion. In the lingual area, a short extension from the distal lower right second tooth to the mesial lower right sixth tooth with a hard consistency was observed  and osteosarcoma were respectively considered in the differential diagnosis.The patient was referred to a maxillofacial surgeon for an incisional biopsy. The biopsy from the right mandibular buccal vestibule was sent to the laboratory in the form of two soft tissue pieces with an elastic consistency and a total size of 20 × 5 × 15 mm. Malignant proliferation of mesenchymal cells with osteoid production was observed microscopically by an oral and maxillofacial pathologist. The cells had pleomorphism, hyperchromatism, mitosis, and bizarre nuclear and cytoplasmic shapes. Osteoid and, in some areas, chondroid matrix were observed within the cells. Eventually, a chondroblastic osteosarcoma lesion was diagnosed histopathologically Figure .The decision was therefore made in favor of completely removing the lesion by an oral surgeon followed by chemotherapy. Upon removal of the lesion, a large 2 × 4 × 6 cm piece was sent to the lab along with the lower right third to fifth teeth. In the microscopic appearance, episodes of neoplastic sarcoma consisting of spindle to round cell proliferation with moderate to severe cytonuclear atypia and atypical mitosis with osteoid and chondroid formation were observed by the general pathologist. Again, it was diagnosed histopathologically as a high‐grade sarcoma consistent with osteosarcoma.After confirming the diagnosis of osteosarcoma in completely removing the lesion, the patient was referred to an oncologist. Lung CT scan, bone nuclear scan and orbital scan were performed. A 6 mm diameter lytic lesion in the superior lateral bone of the left orbital bone was observed on orbital scan by an interventional radiologist who diagnosed the lesion in favor of metastasis. On the recommendation of the oncologist, 12 chemotherapy sessions were prescribed and the patient was then referred to an oral surgeon for mandibular resection. The patient successfully completed the chemotherapy sessions and did not require mandibular resection after assessment by PET scan and only underwent a 3‐month follow‐up.3Osteosarcoma is a highly malignant tumor with a high potential for destruction. It often occurs in the second decade of life, although it also occurs after the age of 50. Studies report different mean ages for onset of osteosarcoma. About 80% of osteosarcoma cases are of internal bone origin and only 6%–8% occur in the orofacial bones, making it a relatively rare malignancy. Nevertheless, the most common sarcoma occurs in the jaw followed by chondrosarcoma.Osteosarcoma has a number of radiographic appearances, including radiopaque, completely radiolucent with ill‐defined border, and mixed. The classical periosteal reaction sunburst appearance can also be observed in osteosarcoma of the jaw, and in some cases, Codman triangle (triangular elevation of the periosteum), spiking root resorption, Garrington sign, and PDL widening are also noticeable.In the study by Fernandes et al., among 11 cases of osteosarcoma of the jaw, five cases had PDL widening and six cases had a sunburst appearance.We did not notice a clear sunburst appearance in our patient Figure . In factBecause some reactive lesions are aggressive, they can be differentially diagnosed as malignant lesions. One such fast‐growing reactive lesion is aggressive CGCG, which in our case was considered as the first differential diagnosis. According to the study by Najar Karimie et al. in 2020, aggressive CGCG lesions mostly occur in women under 30 years of age and in the anterior mandible. Moreover, due to the high growth rate, severe swelling, pain, tooth loosening, root resorption, and cortex perforation, they can be mistaken for a malignant lesion. All such characteristics were similar to our case.In their 2002 review, Stave et al. evaluated 232 cases of CGCG, most of which had an unclear and multilocular border. Additionally and consistent with our case, most cases had bone pain, expansion and tooth displacement.Jeiraj et al. in 2019 addressed some characteristics of aggressive CGCG in distinguishing it from bone malignancies. Age below 30 years, tendency to the front of the mandible, visible bluish lesion behind the mucosa, and blood aspiration in FNA can help distinguish CGCG from malignant lesions such as osteosarcoma. In our case, in addition to the location of the lesion, which was in front of the mandibular molars and similar to CGCG, the absence of three other characteristics could facilitate the definitive diagnosis.On the other hand, early‐stage osteosarcoma can be mistaken for other benign lesions. In their case report, Arnold et al. concluded in 2020 that osteosarcoma can be easily confused with benign fibrous lesions such as fibrosing dysplasia, cemento‐osseous dysplasia and ossifying fibroma, especially in cases of low‐grade osteosarcoma where osteoid is barely visible on histopathological appearance.Because of the potential clinical and radiographic similarities between osteosarcoma and reactive lesions, clinicians should exercise caution in diagnosis and adopt an appropriate and timely approach. The decisions should be made as a team including Oral and maxillofacial medicine specialists, Oral and maxillofacial radiologists and pathologists, Oral and maxillofacial surgeons.Atessa Pakfetrat: Conceptualization; project administration; supervision; visualization. Toktam Zamani: Conceptualization; project administration; writing – original draft; writing – review and editing. Najmeh Anbiaee: Data curation; methodology; supervision. Majid Mirhashemi: Methodology; supervision. Elahe Vazavandi: Data curation; writing – original draft.None.None.I confirm that written patient consent has been signed and collected in accordance with the journal's patient consent policy.


Visceral hemangiosarcomas (HSA) are rare in cats and typically associated with aggressive biologic behavior and poor prognosis. A 4‐year‐old male neutered domestic shorthair cat was presented with a 3‐month history of hematuria and stranguria; ultrasonography identified a large bladder mass. Complete excision was achieved by partial cystectomy. Histopathology and immunohistochemistry for von Willebrand factor confirmed HSA. The cat was treated using adjuvant cyclophosphamide, thalidomide, and meloxicam for 8 months. Abdominal ultrasonography repeated at 2 months and computed tomography repeated at 5 and 19 months after diagnosis showed no evidence of local recurrence or metastasis. The cat was alive at last follow‐up (896 days). Although the cat described in this report experienced a more favorable prognosis compared to other visceral HSA locations, additional cases are needed to further understand the biological behavior of bladder HSAs and guide treatment decisions. HSAhemangiosarcomaRIreference intervalvWFvon Willebrand factor1Urinary bladder neoplasia is rare in cats with an overall reported incidence of 0.07% to 0.18%, with urothelial cell carcinoma being most common.Urinary bladder HSA has been reported very rarely in cats.2A 4‐year‐old, 6 kg, neutered male domestic shorthair cat was referred to the oncology service of the Royal Veterinary College, United Kingdom, for evaluation of a large urinary bladder mass. Hematuria and pollakiuria first had been noted 3 months before referral. Physical examination at that time had identified a thickened bladder on palpation, but was otherwise unremarkable. Symptomatic treatment with meloxicam (0.05 mg/kg PO q24h) and amoxicillin‐clavulanate (8.75 mg/kg PO q12h) was initiated for 10 days. No improvement was seen and stranguria also developed. The dosage of amoxicillin‐clavulanate was increased to 16.75 mg/kg PO q12h for 7 more days, and the urinary supplement Cystophan  also was initiated (2 capsules PO q24h). No further improvement was reported. An abdominal ultrasound examination was performed and identified a diffusely thickened bladder wall at the cranial pole of the bladder, with a heterogenous mass occupying the majority of the bladder lumen (3.4 × 2.5 cm). The cat was referred for further investigation.12/L ; hemoglobin 5.90 g/dL (RI 8‐15 g/dL); mean corpuscular hemoglobin concentration 29.6 g/dL ; mean corpuscular volume 50.8 fL , and reticulocytes 0 × 1012/L. Serum biochemistry results were within normal limits. Because of the presence of a heart murmur, echocardiography was performed and identified hypertrophic cardiomyopathy with no evidence of left atrial enlargement.Upon referral, physical examination identified a grade III/VI left systolic heart murmur and a large palpable bladder mass, but was otherwise unremarkable. Hematology disclosed a mildly hypochromic anemia with slight evidence of regeneration ; red blood cell count 3.93 × 10Streptococcus lutetiensis, sensitive to penicillin, ampicillin, and chloramphenicol. Cytology of the mass identified low numbers of epithelial cells with minimal atypia, but overall was inconclusive because of the low number of nucleated cells. Based on imaging features such as the smooth luminal surface of the mass and irregularity of the serosal margin, a smooth muscle tumor such as a leiomyoma or leiomyosarcoma was considered; other differential diagnoses included transitional cell carcinoma, rhabdomyosarcoma, fibrosarcoma or lymphoma.Abdominal ultrasound examination identified a smoothly marginated rounded mass within the bladder, measuring 4.1 × 2.2 cm and occupying 80% of the lumen  was initiated before surgery but did not eliminate the urinary tract infection despite dose escalation. Because the mass was likely a nidus for infection, surgery was performed without resolution of the infection. At surgery a large, brown‐colored mass originating from the dorsocranial bladder wall was visualized  were observed. Within the neoplasm, approximately 70% of the section examined was affected by vascular thrombosis and coagulative necrosis. Histopathology was indicative of an HSA that was completely excised with narrow (0.7 mm) margins. The diagnosis was confirmed using immunohistochemistry for von Willebrand factor (vWF), which demonstrated positive immunolabeling of the neoplastic cells.Histopathological examination Figure  indicate2 [5 mg] PO q24h), thalidomide (1.7 mg/kg [10 mg] PO q24h) and meloxicam (0.05 mg/kg PO q24h) was initiated 18 days after surgery. Treatment was planned for a duration of 6 months. Hematology and serum biochemistry were performed 2 weeks after starting treatment, with no abnormalities reported and resolution of the previously noted anemia.Adjuvant treatment using PO metronomic cyclophosphamide , after which further treatment was no longer possible because of owner financial limitations. Repeat staging again was performed 19 months after initial diagnosis. At this time, computed tomography of the thorax and abdomen and focal bladder ultrasonography did not identify any evidence of local recurrence or metastasis. Further restaging beyond this timepoint was declined by the owner. The cat was still alive and well at the time of last follow‐up (896 days), based on email communication with the owner.3To achieve a definitive diagnosis in this case histopathology and immunohistochemistry were required. Initial urine cytology, based on a sample collected by catheter suction, was non‐diagnostic because of low nucleated cell numbers. Previous studies indicate the detection of neoplastic cells in only 30% of urinalysis samples from dogs with lower urinary tract neoplasia, but the sensitivity and specificity of different collection methods have not been evaluated.Regarding treatment, although limited information is available to assess the utility of adjuvant chemotherapy for visceral HSA in cats, adjuvant single‐agent doxorubicin chemotherapy initially was recommended in the present case based on the aggressive nature of other visceral forms of HSA in cats and the known survival benefit of adjuvant doxorubicin‐based chemotherapy in dogs with visceral HSA.The cat of our report tolerated treatment with cyclophosphamide, thalidomide, and meloxicam well with no reported clinical, hematologic, or biochemical adverse effects, which is consistent with previous reports.No regional or distant metastases were detected in the present case, either at diagnosis or during post‐surgical follow‐up. Although the biologic behavior of bladder HSA in cats cannot be determined based on a single report, this observation could suggest a less aggressive clinical course compared to other visceral locations because, at the time of diagnosis, metastatic rates of 67% to 77% have been reported for other cats with HSA, with abdominal lymph nodes (33%) and liver (27%) representing the most common locations.The prognosis for bladder HSA in dogs and cats is difficult to assess given the rarity of the disease with only 5 cases previously described in dogs, and only a single histopathologic study reporting bladder HSA in a cat.In conclusion, we successfully treated a bladder HSA in a cat using partial cystectomy and adjuvant metronomic chemotherapy with a good long‐term outcome. Bladder HSA in cats may have a more favorable prognosis compared to other visceral locations, but review of additional cases is needed to further understand the biological behavior of bladder HSA in cats and guide treatment decisions.Authors declare no conflict of interest.Authors declare no off‐label use of antimicrobials.Authors declare no IACUC or other approval was needed.Authors declare human ethics approval was not needed for this study.


Intussusception, or telescoping of the bowel, is a rare condition in the adult population that can lead to serious complications, such as obstruction or ischemia. Most cases of intussusception are idiopathic and present with a pathognomonic “target sign” on imaging. Rarely, in adults, intussusceptions can be found with lead points, some of which may be neoplastic. Treatments for intussusception include air enemas or surgical intervention if enemas are unsuccessful in resolving the telescoped bowel. This case report discusses an atypical presentation of intussusception in an adult female with a “whirlpool sign” on imaging rather than the typical “target sign.” She was found to have incorporation of mesenteric fat into telescoping bowel causing edema and partial bowel obstruction. The affected bowel was removed laparoscopically, and an end-to-end anastomosis was formed. Pathology of the resected bowel revealed a non-immunoreactive inflammatory myofibroblastic neoplasm as the lead point. Most inflammatory myofibroblastic tumors stain positive for desmin, smooth muscle actin, and anaplastic lymphoma kinase (ALK), whereas this patient was non-immunoreactive. The patient tolerated surgery well and is now pain-free with normal gastrointestinal function. This case report hopes to heighten awareness of atypical presentations of intussusceptions, the use of imaging to help aid in uncertain diagnoses, and the appropriate surgical treatment for symptomatic patients. Intussusception occurs when a proximal segment of bowel telescopes into a distal segment. It accounts for 1% of small bowel obstructions in adults, and currently 95% of diagnosed cases are in the pediatric population. The underlying etiology is often idiopathic in the pediatric population; however, a lead point is found in most cases of intussusception in adults [Inflammatory myofibroblastic tumors are uncommon neoplasms that, when found, are typically seen in the adolescent and pediatric populations. These neoplasms are often localized to the abdominal cavity within the omentum and mesentery . We presA 60-year-old Caucasian female presented to the emergency department with the chief complaint of abdominal pain for the past four days. The pain was described as sharp, constant, and worsening over the past 12 hours. The pain was periumbilical with radiation to her lumbar region. There was anorexia, nausea, and two episodes of non-bilious emesis. The patient reports a bowel movement earlier in the day and frequent eructation. Past medical history included gastritis, hypertension, and GERD. Prior surgical history was notable for tubal ligation. The patient reported a partial small bowel obstruction that was managed medically seven months prior. Current medications include simethicone, omeprazole, carvedilol, and verapamil daily.Physical examination revealed a middle-aged obese female with voluntary guarding. The laboratory evaluation was unremarkable. Abdominal CT without contrast revealed dilated small bowel in the mid-abdomen with areas of narrowing Figures -1C. The After discussing risks, benefits, and alternatives with the patient, a diagnostic laparoscopy with possible conversion to open laparotomy was scheduled. Visualization of the small bowel revealed an intussusception in the distal jejunum extending into the proximal ileum Figures , 2B. A hThe removed bowel measured 34 cm and was sent for pathology. The Pathology report revealed a 6.5 cm mass that demonstrated spindle cell proliferation with mixed inflammatory cells. The neoplasm was non-reactive to the following: CD117, DOG1, STAT6, beta-catenin, anaplastic lymphoma kinase-1 (ALK-1), desmin, muscle actin, S-100, and CD34. The diagnosis is an inflammatory myofibroblastic tumor.Intussusception is a common cause of small bowel obstructions in the pediatric population. However, it is rarely seen in adults, with the diagnosis of intussusception being made in 1% of patients with an underlying small bowel obstruction . In contComputerized tomography (CT) and ultrasound (US) are commonly used to aid in the diagnosis. “Target sign” is pathognomonic for intussusception on either US or CT . In thisMost cases of intussusception in adults are due to a lead point. The lead point can be benign or malignant. The lead point in this patient was an inflammatory myofibroblastic tumor (IMT). A total of 80-90% of cases of inflammatory myofibroblastic tumors stain positive for desmin and smooth muscle actin . Other cThis was a challenging case that was confounded by multiple factors. First, the patient was seen by providers at different facilities with non-communicating electronic medical records. Second, the most common radiographic findings associated with intussusception were obscured due to the incorporation of mesenteric fat. Finally, the neoplasm fit the description of an inflammatory myofibroblastic tumor but failed to stain positive for the most common immunohistochemical markers.Intussusception is a challenging diagnosis based on its varied presentation. This case is meant to bring attention to a unique variation. The case here was distinctive because the patient was outside of the typical age range for the diagnosis of both intussusception and IMT. The patient experienced two episodes over one year before receiving definitive care. Radiographically, a whirlpool sign was found instead of the classic target sign. Finally, the lead point was determined to be a non-immunoreactive inflammatory myofibroblastic neoplasm.


Spontaneous idiopathic testicular hemorrhage is an extremely rare entity with few published reports in the literature.We report a case of a 15-year-old boy who had been experiencing intense, left scrotal pain for the previous twelve hours. No previous history of trauma or bleeding disorders. The left testis was enlarged and tender. Left orchiectomy was performed. The entire testis was dusty and dark grossly. Microscopic sections show diffuse intratesticular bleeding with intact seminiferous tubules and spermatogenesis.Spontaneous idiopathic testicular hemorrhage should be considered when evaluating patients with acute scrotal pain. Clinical and ultrasonographic findings and histopathologic evaluation are mandatory to diagnose it. Acute scrotal pain is one of the urological emergencies that can be attributed to different causes and leads to testicular hemorrhage. The differential diagnosis of testicular hemorrhage is wide including trauma, torsion, infection, coagulopathy, tumors, or, rarely, vasculitis. One of the rare causes of testicular bleeding that should be also taken into consideration is idiopathic spontaneous intratesticular hemorrhage which has no identifiable risk factors –5. This A previously healthy 15-year-old boy presented with acute-onset left testicular pain for 12 h. He was afebrile, denied having any dysuria or hematuria, and denied any history of previous trauma. Physical examination showed a left testicle that was diffusely enlarged without any discrete masses, On palpation, the left testis was tender and swollen. The right testis was normal. Ultrasonography shows heterogeneous enlargement of the left testis with areas of decreased echogenicity. No associated masses are identified. Testicular doppler ultrasonography shows absent blood flow. Surgical exploration was decided urgently to save the testicles.Upon surgical exploration, the whole testis and epididymis were diffusely enlarged and dusty dark Fig. , consequIdiopathic intratesticular hemorrhage is a rare disease that was first described in 1914 . Most paScrotal sonography is the gold standard imaging modality for evaluating patients with testicular pain and swelling, especially since it is fast, simple, and noninvasive. It helps in determining the nature of the swelling, and whether it is solid or cystic . The addMagnetic resonance imaging (MRI) may be used to evaluate an acute scrotum when the results of color Doppler are equivocal. Intratesticular hematomas appear as a homogeneous mass with a moderate to intermediate signal intensity on T1- and T2-weighted sequences. To exclude the possibility of malignant tumors, a dynamic MRI contrast enhancement approach can be applied to distinguish the intratesticular hemorrhage that can be treated conservatively from tumors that require surgery since malignant tumors exhibit contrast enhancement with good diagnostic accuracy .The gross features, absence of blood flow and the black discoloration of the testis in idiopathic intratesticular hemorrhage could resemble the necrotic testicle as in our case, and mandates its removal. However, one of the cases reported the normal color of the involved spermatic cord, which would lessen the likelihood of testicular torsion . AdditioThe final diagnosis for most of the cases was made only after surgical exploration and unilateral orchiectomy , 3, 4, bWe reported a case of idiopathic spontaneous intratesticular hemorrhage which is a rare entity. It is still challenging to diagnose this entity and decide how to save the testis if this is possible. This rare entity should be included in the differential diagnosis of unilateral acute scrotal pain. Clinical, radiologic, and histopathological correlation are mandatory to reach the diagnosis.


Malignant primary rectal melanomas (PRM) are rare tumors. Their diagnosis is frequently delayed as these lesions are often mistaken for benign diseases, resulting in extremely poor overall survival. Histological evaluation with special immunohistochemical (IHC) stains is often indispensable for a definitive diagnosis. The main treatment for this condition involves surgical resection. Adjuvant therapy has also been long recommended. We discuss the case of a 60-year-old woman who presented with changes in bowel habits, anal pain, and perineal burning with no bleeding. A digital rectal examination revealed a nodular mass extending 5 cm from the anal verge. Rectosigmoidoscopy demonstrated an ulcerated polypoid tumor extending 4 cm from the anal verge and over 5 cm into the lower rectum. Biopsy and IHC tests confirmed the diagnosis of rectal melanoma. The patient was successfully managed with surgery followed by external beam radiotherapy and a complete response was achieved after 10 months of follow-up. Since mucosal melanomas (MMs) account for less than 1% of all cases of melanoma, this rare entity can develop on any mucosal surface due to the proliferation of pigment-producing melanocytes. However, the head and neck account for the majority of cases followed by the anorectum, which represents 24% of cases . It mostGiven its anatomic location and nonspecific symptoms, which are frequently confused with other conditions, ARMM can be quite challenging to diagnose at early stages, leading to poor survival rates . Due to We present a case of a 60-year-old female diagnosed with primary rectal melanoma (PRM) who was managed with wide local excision (WLE) followed by adjuvant radiotherapy.A 60-year-old female was admitted with a one-month history of changes in bowel habits, anal pain, and perineal burning with no bleeding. The patient had been a known diabetic for 20 years with high blood pressure and had been under treatment; her family history was negative for colon and/or rectal carcinoma. The abdominal examination was unremarkable. A digital rectal examination revealed a nodular mass extending 5 cm from the anal verge. A full colonoscopy demonstrated an ulcerated polypoid tumor extending over 5 cm, and 4 cm from the anal verge, and involving half the circumference; no synchronous lesions were detected. Histopathological examination (HPE) of the mass revealed nests of heavily pigmented fusiform malignant cells containing melanin pigment, and the diagnosis of malignant melanoma was considered Figure .According to immunohistochemical (IHC) staining, the melanoma markers S-100, Melan-A, and HMB-45 were positive. There was no history of other symptoms or a pigmented skin lesion to suggest another possible primary site. Molecular biology showed a TP53 mutation but no B-type Raf (BRAF) or receptor tyrosine kinase (c-KIT) mutations. Thoracic abdominal pelvic contrast CT revealed a large, irregular, heterogenous rectal mass measuring 5.8 x 5 cm and no evidence of nodal or distant metastases Figure .The patient proceeded to have surgery and underwent WLE; the recovery was unremarkable. Histologically, the sections showed a widely ulcerated anorectal mucosa infiltrated by a melanic process, with infiltration of the submucosa and the presence of perineural invasion. Adjuvant external beam radiotherapy was recommended at a total dose of 48 Gy in 20 fractions of 2.4 Gy, and no other adjuvant therapy was indicated. The patient was seen for a follow-up 10 months after undergoing the procedure. No residual tumor was found on digital rectal and sigmoidoscopic examinations, and the CT scan of the chest, abdomen, and pelvis was clear.Primary MMs were identified for the first time in 1859 by Weber . ARMM isMM differs from cutaneous melanoma in numerous aspects. The pathogenesis of this tumor is generally unknown. According to some reports, this neoplasm has many genetic alterations in intracellular signaling cascades, and the ideal approach to target it is yet unknown . In addiGiven its nonspecific symptoms, ARMM might be challenging to identify. Diagnosis and early treatment are often delayed because these tumors are often confused with benign anorectal diseases such as hemorrhoids and rectal polyps ,12. RectA full colonoscopy is recommended to rule out synchronous lesions and for tissue biopsy. Endorectal ultrasonography, CT, and MRI may be useful in determining tumor size, the existence of regional lymph node metastases, and for systemic staging. Approximately, 61% of patients with ARMM present with regional nodal involvement . Our patNotwithstanding their rarity, MMs have been of significant concern due to their aggressive nature and have a poorer prognosis when compared with cutaneous melanomas, and like most malignancies, early detection is critical for optimizing the chances of survival ,2. Many in view of the rarity of the disease and the lack of randomized controlled trials to devise an optimal treatment, surgery remains the major therapeutic intervention for rectal melanomas ,4. AbdomCurrently, no adjuvant therapy can be considered the standard of care for the treatment of anorectal melanoma. In general, radiotherapy has a limited role in MM. It may be used to achieve local control in patients who are unable to undergo surgical resection, or to improve control after surgery, which is the purpose of the treatment, particularly when resection margins are insufficient. As in our patient, adjuvant radiotherapy has been shown to be effective as a complementary method to WLE in anorectal melanomas, as an alternative to APR in terms of quality of life and decreasing local recurrence ,4. For pThere is currently no standard approach for systemic therapy treatment of resectable MM. Vemurafenib and dabrafenib were developed to inhibit BRAF V600 mutations; adjuvant dabrafenib/trametinib combination therapy is a recommended option for patients with resected stage III or recurrent disease and who harbor a BRAF V600-activating mutation. Based on the results of several studies, the PD-1-directed antibody (nivolumab) should be considered as an adjuvant postoperative treatment option for patients with stage III-IV disease at presentation . For patThe recommended post-treatment surveillance program for patients following treatment for rectal melanoma includes external inspection, palpation of inguinal lymph nodes, digital examination, periodic evaluation by colonoscopy, as well as periodic chest, abdominal, and pelvic CT scans; CT or MRI of the brain at least once a year for life is recommended for all patients with melanoma .PRM is a rare but aggressive tumor with no pathognomonic signs. An early diagnosis is extremely crucial in managing these patients. Due to its rarity, adopting standardized treatment protocols has been challenging. The treatment should be focused on reducing morbidity and optimizing quality of life and function. Surgery remains the main treatment modality, while radiotherapy or systemic therapy are considered other options as adjuvant treatment. The prognosis seems to be bleak, despite all attempts to design a multimodal coordinated strategy.


Editorial on the Research TopicKawasaki disease: an ongoing enigma tangled with the appearance of MIS-CKawasaki disease (KD) continues to present a diagnostic and therapeutic conundrum for pediatrics. More than 50 years after the first publication by Dr. Tomisaku Kawasaki , and decIn early 2020, a novel disease entity was recognized in Italy and Great Britain, presenting a few weeks after acute COVID-19 infection. Pediatricians noticed a constellation of clinical features resembling KD, presenting with fever, conjunctival injection, adenopathy and cardiac involvement in children and adolescents , 4. The In this special issue, we focused on how the diagnosis and management of KD evolved in the era of a novel and similar disease: MIS-C. We gained important insights of the epidemiology of both KD and MIS-C. Kim et al. conducted one of the largest nationwide retrospective studies of KD in Korea, and concluded that besides a still unknown infectious agent, important genetic and environmental factors must play a role in KD. On the other hand, Semeraro et al. demonstrated how the severity of acute COVID-19 infection and the prevalence of MIS-C among hospitalized children changed according to the SARS-CoV-2 variants.Universal mask-wearing in the beginning of the pandemic resulted in a dramatic decrease in the prevalence and slight change in the age-distribution of KD. The diagnostic dilemma of KD and MIS-C, especially in ages 6 and older, provoked the question whether KD presentation might have changed after COVID-19 infection due to a different immune response. Besides realizing the different prevalence of MIS-C and KD among various ethnic groups, You et al. demonstrated that certain laboratory markers, such as albumin, may aid in discerning between the two entities. MIS-C and KD do present with different clinical features, however most of the clinical findings are very similar, and the increasing number of positive COVID serologies among children created an even harder task for pediatricians to correctly diagnose patients.This special issue also contains further analysis of promising new diagnostic markers for KD. Zhong et al. showed that circulating non-coding RNAs, mostly microRNA can be not only diagnostic for acute KD compared to other febrile illnesses, but may differentiate acute and convalescent KD as well as KD with and without coronary artery involvement. Mauro et al. presented a comprehensive review of neurological symptoms and conditions in KD and MIS-C, outlining important similarities and differences, and suggesting the importance of long-term follow-up for appropriate management of neurological sequelae. Another relevant aspect of KD and MIS-C was summarized in the review by Jose and Tierney, reflecting the range of coronary artery involvement and cardiac dysfunction noted in patients with MIS-C, emphasizing the importance of creating a standardized follow-up schedule for this disease entity, similar to what we have for KD.COVID is here to stay, and so is MIS-C, creating an ongoing differential diagnostic problem for clinicians to provide optimal management. Several authors concluded in this issue that novel diagnostic modalities are needed to enhance the differential diagnosis of the two conditions. A powerful diagnostic test would help differentiate borderline cases, late presentations, incomplete or atypical KD, and MIS-C occurring in younger age groups. A proper diagnostic test or biomarker would accurately distinguish the two conditions and would be available for practical clinical use. A powerful diagnostic biomarker, as suggested in a previous special issue on KD in Frontiers in Pediatrics , would b1.finding a sensitive and specific test or algorithm distinguishing KD and MIS-C2.identifying children at risk for cardiac complications during initial diagnosis3.establishing long-term follow-up guidelines for MIS-C, and4.evaluating specific therapies to decrease coronary artery aneurysm in both conditions.In the future, basic science research, translational and clinical studies in KD and MIS-C should concentrate on:


Smooth muscle tumors, known as leiomyomas, are benign and can grow anywhere in the body, but the uterine myometrium is the most common site. Vulvar leiomyomas are uncommon and rare. We report a case of a 25-year-old female P (para) 1 L (live) 1 with a previous normal vaginal delivery, who presented two painless swelling of in vulva for one year. The urethral meatus deviated to the left side. After surgical excision, a histopathological examination was performed that confirmed leiomyoma. The patient was discharged after an uneventful postoperative period. A vulvar leiomyoma tumor is often mistaken for a Bartholin cyst, and it is challenging to distinguish between benign and malignant types of vulvar leiomyomas, making diagnosis extremely challenging. Leiomyomas of the uterus are the most common gynecologic neoplasms frequently encountered in females of reproductive age. Up to 30% of females may develop uterine leiomyoma, benign tumors arising from smooth muscle cells. They can occur in any organ system that contains smooth muscle, including the gastrointestinal tract, the genitourinary tract, and the respiratory system . AlthougSmooth muscle tumors are unusual and sometimes mistaken clinically as Bartholin cysts. The labia majora, blood vessel walls, round ligament, the dartos muscle, and erector pili muscle are thought to be the origin of these tumors. Although extremely rare, valvular leiomyomas have been found in the literature, misdiagnosed as Bartholin cysts . We presA 25-year-old, P1 (para one) L1 (live one) female, came to our outpatient clinic complaining of a painless swelling on her left vulva, which was growing in size. Her menstrual cycles were regular, painless, and not associated with clots. She had no significant medical history or surgical history. There was no history of vaginal discharge, difficulty in micturition, or fever, and her bowel habits were regular. She denied any history of sexual trauma, dyspareunia, or other vulvar pathology and had no significant medical history. On examination, a firm, well-defined, non-tender mass was noted in the left valvar region. She appeared to be in fair physical condition, and her physiological parameters were constant. She was afebrile, and there was no inguinal lymphadenopathy. On local examination, the clitoris appeared normal; the urethral meatus deviated to the left side. The patient was catheterized easily, and the labia majora and labia minora were easily separated. On the left side of the labia minora, the first mass/swelling was observed, 1 cm x 3 cm x 2 cm, and the second mass/swelling observed just above the first was 1 cm x 2 cm x 1 cm, which has keratinization on the anterior surface and was stable, not reducible, and painless  of the pelvis was normal. Two palpable masses in the left labium minora, measuring 2 cm x 3 cm x 2 cm and 1 cm x 2 cm x 1 cm, were observed. These masses were well-circumscribed and hypoechoic and did not show vascularity on Doppler imaging. A provisional diagnosis of a vulvar leiomyoma was made, and the surgical excision of the mass was planned under spinal anesthesia. The incision was made on the anterior surface of the tumor, and then enucleation was done. The mass had a whorl-like appearance on the cut section, and the surgical area was sutured (Figure The postoperative recovery of the patient was uneventful. Histopathological examination of the excised mass confirmed the diagnosis of a leiomyoma. The tumor was composed of interlacing bundles of smooth muscle cells Figure . A limited number of vulvar leiomyomas have been reported in the literature, making them sporadic tumors . They usThe ovaries, bladder, urethra, sinonasal cavities, orbits, and kidneys are unusual locations for leiomyomas . InitialThe patient may have anxiety and cosmetic problems due to the size and appearance of the tumor. When cancer is symptomatic, surgical excision of the tumor is a possible treatment ,9. The lWe present a case of vulvar leiomyoma in a young female. A vulvar leiomyoma tumor is mistaken mainly for Bartholin's cyst, and it is challenging to distinguish between benign and malignant types of vulvar leiomyomas, making the diagnosis extremely difficult. The procedure currently used, excisional biopsy, is the most effective. After therapy, a follow-up is necessary if there is any difficulty in that region.


Brown tumor of the knee is a focal benign cystic lesion of bone. The etiopathogenesis of brown tumor is believed to be abnormal bone metabolism in patients with hyperparathyroidism. We present a case of a 32-year-old male with recurrent knee pain, weakness of the lower limb and a nodular mass on left inferior lobe of thyroid. Timely identification of the underlying cause and localization of lesion(s) is important because the management and prognosis vary based on the etiology. The diagnosis of a brown tumor is the result of the sum of the medical history of patients with clinical, radiographic imaging, histopathological, hematological, and biochemical laboratory investigations. A brown tumor also known as osteitis fibrosa cystica is a unique manifestation of reactive focal bony lesion resulting from rapid osteoclastic activity either due to excessive secretion of parathormone (PTH) or paraneoplastic syndrome. The incidence is less than 5% in primary hyperparathyroidism and is more commonly seen in secondary hyperparathyroidism We present a case of a 32-year-old Indian male admitted to the orthopedics department. The patient had a history of recurrent pain in the right knee for the past 4 months. Mechanical diffuse pain was exacerbated during weight bearing and walking and decreased while resting and subsided on taking analgesics. He was on a Vitamin D3 30,000 IU supplement course for 6 weeks. Pain and lower limb motor weakness were the only musculoskeletal complaints. His other systemic medical history was unremarkable. The range of motion of the knee was normal. He had no history of trauma. On physical examination of the knee, tenderness or edema, or external abnormality was not observed. While a nodule was felt in the inferior lobe of the left thyroid.Laboratory analysis showed an elevated serum calcium level of 7.8 mmol/L  and elevated serum alkaline phosphatase level of 590 IU/L  and a low serum phosphate level of 0.54 mg/L (reference range: 0.77-1.32 mg/L) and low total vitamin D10.6 ng/ml(reference range:21-100). Subsequent testing revealed a high PTH level of 138 pmol/L (reference range: 1.2-5.8 pmol/L). Rheumatoid factor, C- reactive protein and Anti cyclic citrullinated peptide antibody, thyroid and other renal function tests were within the normal range.Due to inconclusive test results and continued pain in the right knee and nodule as palpatory finding, the patient was advised for further imaging investigations.On simple radiographs of the bilateral knee joints, the cortical bone of the right distal end of femur showed well defined osteolytic geographic lesions at the epiphysis of medial femoral condyle extending upto the subarticular region. An eccentric lytic lesion with rim-like margins at the proximal end of the tibia showed cystic changes with decreased bone density and the trabecular bone of the upper end of tibia was thinned while the left knee was normal. A and BFiMagnetic resonance imaging (MRI) was performed on the part showing cystic bone change to determine the bone tumor. A well-defined heterogeneously hyper-intense lesion on Short-TI Inversion Recovery and hypointense on T2W1 with T2W1 hyperintense foci in the epiphyseal region of medial femoral condyle with subarticular extension. Another eccentric multiloculated fluid signal intensity lesion with fluid-fluid levels which are hyper-intense on T2W1 and Short-TI Inversion Recovery noted in the upper end of the tibia with moderate marrow edema was observed . Based oFurthermore, neck ultrasound images shows a solid, hypoechoic, well circumscribed mass with color-Doppler of monopolar vascular pattern was located posteroinferiorly to the lower pole of left thyroid lobe, these ultrasound features were typically suggestive of the parathyroid adenoma  and contTo arrive at a conclusive diagnosis, a histopathological study of the tibial lesion was performed and revealed the presence of a giant cell granuloma with microscopic features of increased osteoclasts activity, hemosiderin pigment deposition and bone resorption suggestive of hyperparathyroidism.Hence, the high level of PTH and parathyroid mass on imaging findings has raised the possibility of a brown tumor of the knee. Later, an opinion was sought from the general surgeon who then advised and performed a left-sided parathyroidectomy. Since the patient was lost to follow-up, subsequent changes in the lesion of the tumor are not known.Clinically brown tumor of the knee in the setting of primary hyperparathyroidism is very rare. There has been only 1 report on a brown tumor of the knee by primary hyperthyroidism in the medical literature In conclusion, physicians should keep in mind brown tumors during daily practice as a differential diagnosis. Timely identification of the underlying cause and localization of lesion(s) is important because the management and prognosis vary based on the etiology and the site of the lesion and in the case of hypercalcemia and radiographic evidence of lytic lesion, primary hyperparathyroidism should always be kept in differential diagnosis and should be looked into once more common causes.None.Not commissioned, externally. Peer reviewed.Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.


Hepatocellular carcinoma is a malignancy with an increasing incidence worldwide and is one of the most serious cancers in adults. We encountered a case of initially unresectable massive hepatocellular carcinoma in which conversion to curative resection and pathological complete response were achieved after atezolizumab plus bevacizumab therapy. Atezolizumab plus bevacizumab combination chemotherapy may be one of the most promising options for unresectable hepatocellular carcinoma. The incidence of hepatocellular carcinoma (HCC), the sixth most frequent cancer worldwide, is rising. The best curative choices for locoregional HCC remain resection and transplant. Regarding advanced HCC, new advancements in therapies such as programmed death-ligand 1 (PD-L1) inhibitors and antibodies for the vascular endothelial growth factor receptor (VEGF) have altered management [The combination of the PD-L1 monoclonal antibody atezolizumab and the anti-VEGF monoclonal antibody bevacizumab became the first-line treatment for unresectable HCC . FurtherRegarding surgery, robotic liver resections have broadened their indications and gained acceptance lately ,6. When An 85-year-old man was referred to our hospital for treatment of HCC. He had chronic liver disease due to hepatitis B virus and hepatitis C virus infections. An enhanced computed tomography (CT) scan showed a liver tumor at 82 × 78 mm in the right hepatic lobe in broad contact with the inferior vena cava and the root of the right and middle hepatic veins Figure . InitialWe considered the current tumor unsuitable for resection due to its size and location and immediately introduced the patient to combination biological therapy with atezolizumab 1,200 mg) plus bevacizumab (10 mg/kg). After the second course of biological therapy, his tumor marker levels were within the normal range (AFP: 2.3 ng/mL and PIVKA-II: 19.7 mAU/mL). Courses one to five were managed with atezolizumab plus bevacizumab, and atezolizumab monotherapy without bevacizumab was administered during the following four courses due to proteinuria. His tumor marker levels after the ninth course remained within the normal range (AFP: 1.8 ng/mL and PIVKA-II: 28 mAU/mL). A follow-up CT scan showed a remarkable reduction of the tumor size to 48 × 39 mm after the ninth course . CT volumetry showed a liver volume of 890 mL and a residual liver volume of 846 mL in the case of anatomical resection of the S8 ventral area. During the operation, we found that the clearly demarcated hepatic tumor was located adjacent to both the middle and right hepatic veins but was able to be detached from them relatively easily. Using the saline-linked monopolar scissors (SLiC-Scissors) method, a previously reported novel technique for robotic liver parenchymal transection [The postoperative pathological examination revealed R0 resection and pathological complete response Figure . The recPatients with localized HCC may be cured by surgery, ablation, or liver transplantation. On the other hand, the prognosis for HCC with metastases and locally advanced HCC was four to seven months before the invention of sorafenib . As a muIn our case, an enhanced CT scan at the time of the first visit showed extensive contact with the inferior vena cava and the right and middle hepatic veins, and curative-intent resection required the extended right lobectomy with combined resection of the middle hepatic vein; therefore, we thought upfront surgical resection was not suitable. Because it is strongly recommended by the 2021 Japanese Guidelines on Liver Cancer Examination and Treatment that atezolizumab plus bevacizumab combination therapy should be used as the first-line therapy for unresectable HCC due to its effect of extended survival more than sorafenib [Regarding surgery, since the first laparoscopic liver resection was reported in 1991, it has spread rapidly worldwide. Oncological outcomes from minimally invasive liver surgery have been reported to be equivalent to those from open surgery. Minimally invasive surgery has advantages over open surgery for postoperative pain, intraoperative blood loss, postoperative complication rate, length of hospital stays, and quality of life . ConcernIn this case, we were able to safely dissect the tumor from the right and middle hepatic veins, taking advantage of the superiority of robotic surgery. Thus, robotic liver resection after intensive biological therapy using atezolizumab and bevacizumab may be one of the promising options for unresectable HCC. However, a large number of HCC patients will be required to investigate the characteristics of cases in which atezolizumab plus bevacizumab combination chemotherapy is remarkably effective because this is a single case report.We report our experience with the robotic S8vent anatomical liver resection after atezolizumab plus bevacizumab combination therapy for initially unresectable HCC. The postoperative pathological examination revealed that a pathologically complete response was achieved by biological therapy, and the patient is doing well and free from oncological therapy. Surgical resection after atezolizumab plus bevacizumab combination therapy may be one of the most promising options for the treatment of unresectable HCC.


Primary paratesticular liposarcoma is rarely diagnosed among urinary tumors. In this study, through the retrospective analysis of clinical data and literature review, a case of recurrent paratesticular liposarcoma with lymph node metastasis after radical resection has been reported to explore novel strategies for the diagnosis, treatment and prognosis of this rare disease.The present case involved a patient who was misdiagnosed as a left inguinal hernia for the first time two years ago, but was later diagnosed as mixed liposarcoma by using postoperative pathology. Currently, he is readmitted to the hospital with a recurrence of the left scrotal mass for more than 1 year. Combined with the patient's past medical history, we performed radical resection of the left inguinal and scrotal tumors and lymphadenectomy of left femoral vein. The postoperative pathology indicated that well-differentiated liposarcoma was accompanied by mucinous liposarcoma (about 20%), and lymph node metastasis of left femoral vein both of which occurred at the same time. After the operation, we recommended the patient to receive further radiation therapy, but the patient and his family refused, hence we followed up the patient closely for a long time. During the recent follow-up, the patient reported no complaints of discomfort, and no recurrence of mass in the left scrotum and groin area.After conducting extensive review of literature, we conclude that radical resection remains the key to treat primary paratesticular liposarcoma, while the significance of the lymph node metastasis is still unclear. The potential effects of postoperative adjuvant therapy depends on the pathological type, and hence close follow-up observation is essential. Sarcomas account for approximately 15% of all the childhood malignancies, compared with 1% of all adult malignancies. The most common subtypes of soft tissue sarcoma (STS) include undifferentiated pleomorphic sarcoma (UPS), gastrointestinal stromal tumors (GIST), liposarcoma (LPS) and leiomyosarcoma (LMS). Liposarcoma is mainly composed of mesenchymal cells formed during the process of differentiation from adipoblast to adipocyte, ranking second among the soft tissue sarcomas, and accounting for about 15%–20% . ParatesAt present, there is little clinical understanding about the clinical characteristics, diagnosis, treatment and prognosis of PLS. In this article, we report a case of recurrent left PLS in patient, who was misdiagnosed as indirect inguinal hernia entering the scrotum at the first presentation, but then relapsed and was admitted to the hospital 2 years after the surgery. In addition, combined with the related treatment experience and comprehensive literature review, we have attempted to clarify the clinical diagnosis and treatment of this rare disease.A 72-year-old Chinese man was admitted to our hospital again for more than one year because of the recurrence of the tumor in the left scrotum and groin area.3 years ago, the patient accidentally discovered a lump in his left groin area, which was about 4 cm × 5 cm in size at that time. No attention was paid to consult a doctor, and then it gradually enlarged and entered the scrotum, with a size of about 20 cm × 20 cm. The surface was not found to be swollen and tender, so it could not be absorbed into the abdominal cavity. 2 years ago, the patient first visited the general surgery department of our hospital. At that time, combined with B-mode ultrasound, he was diagnosed with indirect inguinal hernia, and agreed to undergo hernia repair for the patient. During the operation, there was no hernia sac bulge, but a solid tumor with the appearance of adipose tissue was found in the scrotum, most of which were soft, some of which were tough, yellow and wrapped around the testicles and epididymis. Therefore, we performed scrotal tumor resection for patients, completely removed the tumor base, spermatic cord as well as the testicular tissue, and performed high ligation of spermatic cord. It was extremely difficult to diagnose fast freezing pathology during operation. Postoperative routine pathology showed mixed liposarcoma, mainly WDLPS, accompanied by MLPS and DDLPS. The tumor size was 22 cm × 14 cm × 8 cm, and the blood vessels were rich in the tissues, which in the focal area showed tumor-like hyperplasia. The capsule was complete, and the surgical margin was observed to be negative. Immunohistochemistry revealed Ki-67(5%), P53(+), S-100(1+), CD68(−), CD34(−), SMA(−), PCNA(3+), CD56(−). Patients and their families refused further adjuvant treatment after the surgery. More than a year ago, the tumor in the left scrotum and groin area recurred. At present, he is admitted to the urology department of our hospital. Except the swelling of scrotum, he has no other major complaints.The patient has hypertension for more than three years and is taking hydrochlorothiazide once a day for the same. He is also suffering from tipe 2 diabetes mellitus for laat three years and is on acarbose 50 mg once a day and glimepiride 1 mg once a day. The patient did not have any other surgical history.The patient did not have any addiction to tobacco and alcohol. And he has no genetic history of cancer.A lump of about 25 cm × 20 cm in size could be felt in the left scrotum and groin area, with tough texture and clear boundaries .After admission, the blood routine, coagulation function, liver as well as kidney function, tumor index and other blood biochemical tests of the patients showed no obvious abnormality.Two years ago, when the patient first visited a doctor in general surgery, abdominal enhancement computed tomography (CT) showed that there was a lumpy uneven density shadow in the left scrotum, and there was fat and soft tissue density shadow in it, which was not related with the contents of the abdominal cavity. After enhancement, some nodules were slightly enhanced, but the remaining enhancement was not found to be obvious. Soft tissue density nodules were found in the right groin area, but the enhancement was not obvious, and no enlarged lymph nodes were detected retroperitoneum . After tConsidering the recurrence of liposarcoma, radical tumor resection was performed. During the surgery, a tumor with a size of about 20 cm × 18 cm in the groin and scrotum was found, with a tough texture and a cystic mass, which was adhered to the surrounding tissues. After electrocoagulation with Ligsaure and separation, the tumor was completely removed . At the There were no related complications in the patient after the surgery. We recommended that the patient should undergo further radiotherapy, but the patient and his family still refused. Therefore, we could only follow up the patient closely. At present, up to more than one month before submission of the manuscript, the last reexamination of the patient showed that there was no recurrence of the lump in the scrotum and groin area, and CT scan exhibited no regional soft tissue density shadow. Further follow-up is in progress currently.Sarcoma of genitourinary system is extremely rare malignancy, accounting for only 1%–2% of malignant tumors of genitourinary system. Most of them are diagnosed in children, and only a few cases are found in adults , 8. AmonPLS usually presents itself as a slow-growing lump in the groin or scrotum, and the lump often grows progressively without pain and discomfort –16. TherLaboratory biochemical tests lack specificity in the diagnosis of PLS. Ultrasound is usually the first choice for imaging examination of the soft tissue tumors, and it plays a significant role in differentiating the cyst, hydrocele and epididymitis. PLS ultrasound usually shows solid, hyperechoic and heterogeneous lesions, but for the large tumors, identification of the origin and boundary of the tumor is often limited . Computein situ after operation is significantly high (about 50%)   and someout 50%) , 28. DDLout 50%) . The locout 50%)  and a moThorough surgery to obtain the negative surgical margin is the common strategy for the treatment of histiocytoma at present. For PLS, the widely used surgical methods are radical orchiectomy with extensive local excision and high spermatic cord ligation . IncomplAt present, there is still controversy about the clinical effect of adjuvant radiotherapy or chemotherapy in PLS patients. Most scholars believe that preoperative or postoperative radiotherapy and chemotherapy are unable to substantially reduce the recurrence rate of WDLPS, and radiotherapy has no significance for localized lesions , 40, andp = 0.010), tumor size , dedifferentiation and round cell subtype  were found to act as independent risk factors responsible for the tumor recurrence.The prognosis of patients mainly depends on the tumor site, tumor size, pathological type, classification and staging, and the degree of surgical resection. The tumor location is the most important factor for determining the prognosis. It has been reported in the literature that the tumor is primarily located in the retroperitoneum relative to the superficial parts such as limbs, and the risk of recurrence and metastasis is higher. PLS tumor diameter is an important factor for determining the success of radical surgical resection , especiaIn summary, PLS is a rare tumor, which predominantly presents itself as irregular masses with different sizes and medium-soft texture in the scrotum, spermatic cord or groin in clinical. The laboratory tests are mostly negative, as well as heterogeneous and solid lesions can be observed based on imaging examinations. It can be easily misdiagnosed before the surgery, and careful physical examination is necessary. The possibility of PLS should also be considered in the identification of the fat-containing inguinal scrotal masses. The imaging procedures such as Ultrasound, CT and MRI can provide relevant imaging information on the nature of pre-operative liposarcoma. Immunohistochemistry and molecular testing of MDM2 and CDK4 can contribute to an accurate diagnosis. Therefore, based on the literature review, we suggest that in the case of rapid intraoperative cryosurgery with a definite pathology, extensive local excision and radical orchiectomy with high ligation of the spermatic cord in order to obtain a negative margin should be conducted as the best therapeutic strategy to maximize the chance of surgical cure. Incomplete resection can result in high local recurrence rate and shortened recurrence time after PLS resection. The histopathological type and malignant grade could be helpful to judge the prognosis, and further adjuvant treatment should be selected according to the different pathological types. Novel targeted therapies may be promising, but the long-term close patient follow-up is essential for optimal treatment.


Benign adnexal neoplasms are quite a common occurrence in adults, especially in the head and neck region. They raise suspicion for malignancy if there are red flag signs like rapid increase in size, pain, ulceration or recurrence. We hereby report a case of a middle-aged gentleman who consulted our surgical OPD with right-sided neck swelling, which was initially thought to be a dermoid cyst; on further evaluation, found to be a dermal sweat gland tumour with features of nodular hidradenoma. The point that is of interest but coincidence to note is that this swelling was preceded by a minor trauma.Characterising these swellings using simple imaging and pathological investigation modalities is important to study their behavioural pattern and add the same to our existing database. This will also help the treating surgeons to keep in mind the possibility of occurrence of such histologies in soft tissue swellings when they present with uncommon clinical features, instead of brushing them aside as the common epidermal or dermoid cysts. Incidence of malignancy is almost nil in nodular hidradenoma, which when found, is attributed to poor surgical clearance; hence the prudence to operate with adequate clearance is extremely significant in preventing the transformation of a mole into a mountain. Nodular hidradenomas or clear cell hidradenomas are asymptomatic, benign, nodular intradermal tumours that make up 95% of all hidradenomas . AlthougThey usually present as well-defined and multilobulated tumours seen only in the dermis or in both the dermis and the subcutaneous tissue. The primary pattern is mostly solid, the other is both solid and cystic, and occasionally tubulocystic structures are observed. These tumours are slow-growing and are usually present for many years before diagnosis .In rare cases, malignant transformation of clear cell hidradenoma with metastatic spread has been reported .The point of interest but coincidence in our case is that the swelling was preceded by a minor trauma. Owing to the patient's clinical signs and symptoms and history, a preliminary diagnosis of implantation dermoid cyst was made. But with further investigation, it was found to be nodular hidradenoma.A 48-year-old man presented with a swelling on the right side of his neck that had been present for the past two months. The patient reported that the swelling developed after a pin prick injury. Upon examination, a firm, smooth, and freely mobile swelling measuring 3x3 cm was observed on the right posterior triangle of the neck. The swelling was firm, non-tender with a smooth surface; had a fluctuant summit; freely mobile and the skin overlying the swelling was pinch-able except over the summit Figure . Based oHidradenomas are benign skin adnexal neoplasms that are rare and encompass a wide range of differentiation and most commonly occur in adults. The exact incidence rates of nodular hidradenomas have not been characterized; malignancy is reported to be less than 0.001% in various studies. They present as solitary tumours that are generally firm but freely mobile . They arThe histology of a nodular hidradenoma can be described as a lobulated mass that is well circumscribed which mainly extends to the dermis and sometimes, can spread subcutaneously . Two celThe tumour cells generally express AE1/AE3, EMA, and CEA . A transIt is generally a slow-growing tumour. On the other hand, if it progresses quickly, it may be a sign of malignancy . Since, The precise incidence rate of transformation to malignant hidradenoma has yet to be definitively established. However, it remains an elusive figure within the current body of knowledge. Notably, malignant hidradenomas exhibit an elevated expression of PHH3 greater than 0.7% and/or Ki-67 exceeding 11%. These markers serve as indicators of increased cellular proliferation and activity, further underscoring the complex nature of malignant transformation in hidradenoma [Emphasising the significance of comprehensive surgical intervention with adequate margins is paramount in mitigating the risk of local recurrence. The optimum margin is when the margins are clear of the tumour both macroscopically and microscopically. Wide surgical excision, encompassing an appropriate margin of healthy tissue surrounding the lesion, is crucial in ensuring complete removal of nodular hidradenoma and reducing the likelihood of its resurgence in the local area . By emplNodular hidradenoma recurrence is common, occurring in up to 10% of cases. This high recurrence rate is largely attributed to inadequate excision of the tumour during the initial surgical intervention . Close mNodular hidradenomas, also known as clear cell hidradenomas, are predominantly asymptomatic and benign nodular tumours located within the dermis. They have a slow growth rate and can go undiagnosed for many years. These tumours often remain asymptomatic and may not be noticed until they reach a significant size or cause noticeable changes. Achieving meticulous and comprehensive excision of nodular hidradenoma is of utmost importance in order to minimize the risk of recurrence and optimize long-term patient outcomes.


Solitary plasmacytomas (SPs) are tumors characterized by local monoclonal plasma cell proliferation, presenting without systemic manifestations. It mainly affects the axial skeleton, with calcaneal involvement being extremely rare. We report a case of a 48-year-old patient with a history of gunshot injury to his foot who presented with worsening heel pain and a calcaneal cyst. Biopsy revealed plasmacytoma, and subsequent 18F-fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) scan further supported the diagnosis of solitary plasmacytoma of the bone (SPB). Management included lesion excision, bone cement placement, and radiotherapy. However, due to recurrent osteomyelitis following cement placement, the patient eventually required total calcanectomy. SPB usually affects older adults, and developing the disease at a young age and in the calcaneus is exceedingly uncommon. Trauma is implicated as a possible inciting trigger in the pathogenesis of SPB without a clear association. This case highlights the importance of developing our current understanding of the clinical presentation and manifestations of SPB, beyond the conventional assumption that it only affects the axial skeleton of older individuals. Plasmacytomas are tumors characterized by the proliferation of monoclonal plasma cells in soft tissue or bone, presenting without systemic manifestations . Based oWe report a case of a 48-year-old male with a past medical history significant for a gunshot injury to his right foot, hypertension, and poorly controlled diabetes mellitus. His right hindfoot was injured in 1986, after which the patient started complaining of recurrent foot pain at the same site. This pain was notably exacerbated by cold temperatures and was alleviated by over-the-counter analgesic medications.In 2020, the patient sought medical attention due to increasing pain intensity associated with a 15-kilogram weight loss over the past nine months. An X-ray revealed a lytic lesion at the posterior part of the right calcaneus Figure . The patFollowing cement placement, the patient continued to experience persistent ankle pain due to recurrent attacks of osteomyelitis. As a result, the 18F-FDG PET/CT whole-body scan was repeated. It revealed a focal lucent bone area measuring approximately 4x4x3 cm with internal cement and an associated hypermetabolic sinus tract with an underlying calcaneal bone defect, suggesting chronic osteomyelitis with sinus tract formation. Therefore, the bone cement was removed to eliminate the source of infection and improve the effectiveness of antibiotic therapy.Klebsiella pneumoniae, which was only sensitive to colistin and tigecycline. A new 18F-FDG PET/CT scan showed the persistence of the same lucent bone area with a new medial cortical defect containing fluid and gas densities connected to a lateral skin defect  has established criteria for diagnosing SPB. The criteria include the following: (1) normal skeletal survey and the absence of any other lytic lesions, (2) biopsy-proven solitary tumor of bone with evidence of clonal plasma cells, (3) the presence of plasma cells within the biopsy, and (4) the absence of clonal plasma cells in the BM aspirate. In order to diagnose SPB, other conditions, such as multiple myeloma, must be ruled out by examining the BM, along with ensuring the absence of CRAB manifestations described above . Using CIt is reported that roughly 50% of patients diagnosed with SPB are at risk of developing MM, with the median time of progression being two to three years . Given iWith a control rate of approximately 80%, localized radiotherapy is considered the treatment of choice for SPB . There iPatients diagnosed with SPB typically have a median overall survival of approximately 10 years. 50-60% of these patients progress to develop multiple myeloma, which is possibly explained by the persistence of undetectable tumor cells not lying within the field of radiotherapy . These fWe have described the case of a middle-aged man who presented with SPB in a relatively rare location, most likely due to trauma endured years prior. SP can infrequently arise in the appendicular skeleton of young patients, particularly in those with a history of trauma. The exact significance of the association between SPB at a young age and a history of preceding trauma remains to be established. A biopsy of the lesion with evidence of clonal plasma cells is required for establishing the diagnosis. Moreover, it is fundamental to exclude multiple myeloma before initiating treatment. Localized radiotherapy remains the best treatment modality for SP, and regular follow-up is essential for monitoring disease progression to multiple myeloma and addressing disease recurrence or delayed complications.


Bladder cancer is characterized by spatial and temporal recurrence in the urinary tract. We describe a case of recurrence at a uretero‐ileal anastomosis after radical cystectomy and nephroureterectomy. It was difficult to control bleeding from the tumor, but hemostasis was achieved.A 73‐year‐old man with a history of radical cystectomy and reconstruction of the ileal conduit and right nephroureterectomy was diagnosed with recurrence at the uretero‐ileal anastomosis site. Bleeding from the tumor could not be controlled by flexible gastrointestinal endoscopy. The patient underwent coagulation via an ileal conduit approach using a rigid scope and bipolar electrocautery, which is usually a modality for transurethral resection.This is the first report in which a modality normally used for transurethral resection was used to control bleeding in a patient with an ileal conduit. This application is useful in cases open surgery or additional irradiation might be difficult. We report a case in which we controlled the bleeding from a recurrence tumor at anastomosis using only electrocoagulation via an ileal conduit. In situations where open surgery or additional irradiation are considered difficult, this method might be an option.CTcomputed tomographyNMIBCnon‐muscle‐invasive bladder cancerRCradical cystectomyTURBTtransurethral resection of bladder tumorUCurothelial carcinomaBladder cancer is characterized by spatial and temporal recurrence in the urinary tract. After RC, the rate of recurrence in the urinary tract is 4%–10%.The patient was a 73‐year‐old man with a past medical history of significant diabetes, asthma, hypertension, and percutaneous coronary intervention for coronary stenosis, and he took aspirin. He had a history of three TURBT with pathologically confirmed high‐risk NMIBC followed by RC and reconstruction of an ileal conduit by Wallace plate technique at age 69. Pathological examination revealed a UC, low grade (G2), pTa, and the bilateral margins were negative. Furthermore, he underwent right open nephroureterectomy due to right lower ureteral recurrence at age 72. During the operation, because part of the ureter‐ileal anastomosis was firmly adhered, the ureter was dissected and cut as far as possible distally. Adjuvant chemotherapy was not administered based on the pathological findings of UC, high grade (G2), pT2, and absence of lympho‐vascular invasion and a resection margin.One year after the right nephroureterectomy, CT showed left hydronephrosis and acute pyelonephritis. A recurrent mass at the left uretero‐ileal anastomosis was noted Fig. . After lSix months after irradiation, CT showed multiple lung metastases, lumbar bone metastases, and aortic lymph node metastases, and then systemic chemotherapy with gemcitabine/carboplatin was planned.However, at the time of chemotherapy induction, the patient developed severe anemia with a Hb level of 5.6 g/dL due to ileal conduit bleeding. CT showed re‐enlargement of the recurrent mass at the uretero‐ileal anastomosis Fig. . The patPostoperatively, systemic chemotherapy was introduced, and no rebleeding was observed during the 8‐month follow‐up.et al.We performed nephroureterectomy on the first recurrence of the right lower ureter. No tumor infiltration was observed microscopically at the resection margins, although we were unable to remove the uretero‐ileal anastomosis. Usually, however, the anastomosis should be removed using a technique such as segmental uretero‐ileal conduit resection, as reported by Zeng et al., the anastomosis was the most common site of recurrence after ileal conduit creation, and surgical resection was the treatment of choice in nearly 80% of cases,In this case, the second recurrence may be a recurrence of the residual ureter at the time of nephroureterectomy. In a report of 19 cases by Zattoni When dealing with bleeding in patients with a history of treatment like this one, hemostatic maneuvers similar to those used for gastrointestinal bleeding are an option. Ultimately, we chose a rigid cystoscope, which has two advantages. First, because an ileal conduit is a totally incontinent urinary diversion, the intraductal pressure does not increase even during the procedure requiring irrigation. As a result, rupture or anastomotic leakage can be prevented. Second, the high perfusion efficiency provides a relatively good field of view. This method could be one of the options for dealing with cases such as the present case.We experienced a case of controlled bleeding from a UC recurring at the uretero‐ileal anastomosis, which was treated by electrocoagulation via an ileal conduit. Although this appears to be a safe procedure, further study is needed for cases in which palliative treatment must be selected.Rikuto Yasujima: Conceptualization; writing – original draft. Yasukazu Nakanishi: Conceptualization; supervision; writing – review and editing. Kohei Hirose: Supervision; writing – review and editing. Yosuke Umino: Supervision; writing – review and editing. Naoya Okubo: Supervision; writing – review and editing. Madoka Kataoka: Supervision; writing – review and editing. Shugo Yajima: Conceptualization; supervision; writing – review and editing. Hitoshi Masuda: Conceptualization; supervision; writing – review and editing.The authors declare no conflict of interest.Not applicable.Informed consent was obtained from the patient for the release of this case report.Not applicable.


Talon noir is an asymptomatic, self-limiting, dermatological condition that is most commonly seen in young athletes and mountain climbers. Histologically, the characteristic findings are intraepidermal hemorrhage and fluid collection due to shearing forces. Here, we report an interesting case in an elderly female, which mimicked acral melanoma clinically and alerted us to do a biopsy for confirmation. Talon noir or black heel is a dermatological condition characterized by intraepidermal hemorrhage and exudation due to shearing forces in an area of recurrent or sudden trauma . It is cIn our case, we report a case of talon noir in a middle-aged social activist woman with no history of sports activity.A 49-year-old diabetic female, a radio announcer and social activist by profession, presented with two dark blackish-colored plaques on her right sole for one year. These lesions were painless and gradually increased in size. There was no history of injury or trauma. However, the patient provided a history of using high-heeled shoes and walking long distances for work. On examination, two irregular, thickened, blackish-brown plaques were identified on the plantar aspect of the right forefoot measuring 4.5 × 3 cm and 1.5 × 1.5 cm, respectively Figure . There wTalon noir was first described in 1961 as asymptomatic, bilateral, deep-seated petechiae on posterolateral aspects of the heels of basketball players . There aWe searched the literature on PubMed and Google Scholar using the keywords “Talon noir,” “Black heel,” and “Subcorneal hematoma” from 2012 to 2021 and retrieved 14 articles with full text or abstract available reporting 51 such cases in English literature. Most cases were reported involving the heel, sole of the foot, toes, and palm of adolescents and young adults with a history of sports activities, repeated trauma, mountain climbing, or taking anticoagulant therapy. A few cases were also observed involving extremities of age with no such typical histories. An unusual case of a viral infection-associated black heel has also been reported ,7.Clinically, it presents as asymptomatic, asymmetrical, black-to-brown-colored macules, isolated or in clusters, with an uninterrupted normal skin surface . The patDifferential diagnoses usually include acral melanoma, acral nevi, plantar wart, pyogenic granuloma, angiokeratoma, corn, and tinea nigra ,5. A warDermoscopy, a non-invasive procedure, can differentiate talon noir from melanoma preoperatively as the former shows homogenous reddish globular structures . Acral mIt is a self-limiting benign condition that usually resolves within a few weeks by avoiding sports activity or any kind of repetitive trauma and using well-cushioned shoes, thick socks, or skin lubrication. Exanthema-associated capillary fragility can be benefited from vitamin C supplementation ,11-14. ITable The present case mimicked melanoma due to its dark blackish color and irregular ulcerated surface. However, awareness of this entity could have saved the pain of excisional biopsy and the associated psychological trauma of being diagnosed with cancerous growth albeit provisionally. The importance of detailed history particularly of sports or long-distance trekking cannot be over-emphasized. This case may be a complication of diabetes; nonetheless, future studies are mandated due to the paucity of supportive literature. Thus, pigmented lesions are not always worrisome melanocytic lesions.


A 9-year-old male presented with an asymptomatic, confluent, and hyperpigmented skin lesion on the right lateral trunk region presented at birth. No changes in color and consistency are reported, except that the lesion enlarged in proportion to the patient's growth. The mother informed us that the patient was diagnosed with anxiety one year ago. Physical examination revealed a large, hyperpigmented, and confluent patch of overgrown skin with a dimension between 20cm long and 15cm wide associated with painless verrucous plaques located on the right lateral trunk region (A). The laboratory investigations revealed a H1047R mutation in the PIK3CA gene. The anatomopathological study showed hyperkeratosis, acanthosis, and papillomatosis compatible with verrucous epidermal nevi (hamartomas). Anteroposterior (B) and lateral (C) neck radiographs revealed left convexity scoliosis. Based on the clinical, pathologic findings, the lesion was diagnosed as verrucous epidermal nevus (VEN). This malady is benign and congenital and could be associated with abnormalities in neurologic, ophthalmologic, or skeletal systems. When this type of nevi is located on the trunk, is associated with alterations in the bone curvatures of the spine, and/or in the bones of the arms or legs due to mutations in the PIK3CA gene, as in our case. The treatment was a shave excision followed by a phenol peeling medical; although, it is not possible to predict when skin lesions will recur.