Canine osteosarcoma (OS) or osteogenic sarcoma is an aggressive tumor of the skeletal system, associated with a rapid progression and guarded prognosis. The osteosarcomas, mostly arise from the appendicular skeleton while axial OS (osteosarcoma of flat bones) are less reported in the majority of large breeds. This report describes complete para-clinical investigations of an aggressive chondroblastic OS involving facial flat bones with highly metastatic characterization in a large mix breed stray dog. Radiographic and computed tomography findings demonstrated an amorphous and active new bone formation, associated with the severe lytic areas in the left maxillary, orbital and zygomatic bones. Also, lots of nodular densities were distributed in all lung lobes. The cytological examination of the mass revealed individualized oval to spindle-shaped pleomorphic mesenchymal cells exhibiting many criteria of malignancy such as marked anisocytosis, anisokaryosis, prominent and multiple nucleoli. The punctate cytoplasmic vacuoles were obvious and bi-nucleated cells were frequently observed. These cells were seen in the background of an eosinophilic matrix. Histopathologic evaluation of the mass exhibited areas of osseous differentiation within the mass containing bony spicules and wavy bone formation along with the vast areas of cartilaginous differentiations with chondroblasts in lacunar spaces. Ultimately, chondroblastic OS with severe lung metastasis was confirmed and the animal was euthanized. Fibroblastic OS is found to have a more favorable prognosis than the other subtypes in dogs.2 Differently, chondroblastic OS has a high rate of metastasis. Metastasis to the lungs is more common, but it can occur to the other bones and organs as well.3 The lesion is not generally uniform and emerges as a severe, active and aggressive new bone formations and periosteal reactions. It has been rarely reported from domestic animals, but its characterizations are the same as in humans.4 The most predisposed breeds of dogs are large breeds and the mean age of the involvement is eight years.5 Axial OS is locally aggressive and destroys bone locally, it can invade into surrounding tissues and causes dystrophic calcification.3 Despite advances in various treatments  prognosis is still poor, with median survival time from three months to one year. This case report describes a metastatic form of chondroblastic OS, involving flat bones of the skull with severe lung metastasis in a young dog.Osteosarcoma (OS) is a malignant primary bone tumor defined as a bone matrix-producing neoplasm arising from primitive transformed mesenchymal cells that differentiate into osteoblasts.A young (about 2-years-old) intact male, mixed-breed dog was referred to the Veterinary Teaching Hospital, University of Tehran with a history of weakness, lethargy, and depression, because of presence of a massive swelling with progressive growth around the left eye with subsequently deformity of the left orbital bone and loss of sight. Skin injuries due to the self-trauma in association with purulent secretions have been noticed during the clinical examination . The masStandard digital radiographs using Direct-View, Classic CR System  were taken in lateral oblique and dorsoventral (DV) of the skull and left lateral, right lateral and ventrodorsal (VD) projections of the thoracic cavity . Radiography of the skull revealed severe aggressive, amorphous and active new bone formations around the left zygomatic bone, orbital bone and maxillary sinus associated with regional soft tissue swelling . rd intercostal space) and shape. There was no sign of pleural effusion and pulmonary vessels, trachea, caudal vena cava and aortic diameter were normal subjectively  and acepromazine . During anesthesia, continuous monitoring and observation were performed. The CT-Scan examination was performed using a multi-detectors helical machine  with the following setting: slice thickness: 1.00 mm; tube rotation time: 1 sec; pitch 1; 120.00 kVp; and 110 mAs. In addition, 750 mg kg-1 of iodinated contrast medium  was injected intravenously to obtain post contrast images. After the imaging process, images were reconstructed to a high- spatial- frequency reconstruction algorithm (bone-U90) without additional filters. Furthermore, all images were reconstructed on a 2- dimensional  and 3-dimensional viewing to allow a better understanding of the regional anatomy  from the mass was taken under ultrasound guidance. Doppler study during the ultrasound also showed the high vascularization of the lesion, although the deeper parts could not be evaluated due to far acoustic shadow produced by more superficial lesions. Multiple samples from different parts of the mass were sampled and sent to the laboratory for the cytological evaluations. Finally, the patient received high doses of anesthetic drugs for euthanasia upon the dog's owner requested. During necropsy, adequate amounts of the neoplastic and aggressive bone lesions were sampled for histopathologic examinations.To evaluate the cytological features of the mass, FNA samples and impression smears from the tissue biopsy were prepared and stained with Giemsa. The stained tissue sections were highly cellular. Pleomorphic oval to spindle shaped mesenchymal cells exhibiting many criteria of malignancy such as marked anisocytosis, anisokaryosis, prominent and multiple nucleoli were observed. Punctate cytoplasmic vacuoles  and few Binucleated cells were frequently seen in the tissue sections of the tumor. The nuclei had coarse nuclear chromatin containing multiple prominent nucleoli. The cytological findings confirmed the presence of a sarcoma most likely OS or chondrosarcoma. Histopathology examination showed a mesenchymal proliferation of neoplastic cells in different parts of the tumor. Areas of osseous differentiation within the mass containing bony spicules and wavy bone formation were associated with the vast areas of cartilaginous differentiation with chondroblasts in lacunar spaces . Frequen6 About 90.00% of humans OSs are conventional OS and about 25.00% of them are chondroblastic OSs.7 Patnaik reviewed 14 cases of canine extra-skeletal OS and chondrosarcoma in which osteo-blastic OS were the most common type and there was just one case of chondroblastic OS that chondroid tissue was its predominant component.8 The FNA as a less invasive and less expensive examination9 was performed for this case owing to good sensitivity (86.00%) and specificity (95.00%) of cytology for diagnosis of OS, but the chondroid nature of the masses is challenging to clinical pathologists that requires histopathology examination.7,10 Although a tissue section from cartilaginous areas of the neoplastic mass would suggest an erroneous diagnosis of chondrosarcoma, which in such cases, radiographic findings such as aggressive appearance, lead to the diagnosis of chondroblastic OS.11 Generally, patients with axial skeleton tumor classified as cranial OS have a high rate of metastasis,12 which was observed in the present patient. In addition, the current patient notably showed clinical signs of lethargy, anorexia, weight loss and weakness in daily activities like the previously reported case of axial OS.12Osteosarcoma is the most common neoplasm of all skeletal system tumors in dogs.13 Radiographically, OS presents a variation of lytic, mixed or productive patterns. However, lesions are not pathognomonic, and similar patterns can occur with other bone tumors. Further, radiographic appearance is dynamic and may change over time.13 Similarly, in the present patient, a mixed pattern of irregular lysis and amorphous new bone formation was detected in radio-graphy. Advanced imaging such as CT-Scan and magnetic resonance imaging have been demonstrated to play an important role in evaluating the extent of tumor for surgical planning; this has resulted in improved survival in OS of the mandible and maxilla in humans.14 The CT-Scan is the preferred imaging modality for biopsy planning in the maxilla-facial region. Furthermore, CT-Scan has increased sensitivity for the detection of pulmonary metastatic disease. In CT-Scan (plain and angiography) an aggressive bony proliferation and lysis, associated with a large number of cannon-ball with various sizes were notably visible in the current case. The type of tumor cannot be determined based on imaging diagnosis alone, and biopsy is always required for definitive diagnosis.14 It is important to consider the diagnostic imaging findings in light of clinical features including tumor location and the histopathological findings. The euthanasia was the owner's approach of choice because of the tumor extension into adjacent orbit regions, and also severe lung metastasis making the surgical procedure not viable.Generally, the clinical presentation and various imaging modalities can lead to a presumptive diagnosis of OS, but the final diagnosis ultimately depends on histo-pathologic evaluation.


Extra-uterine manifestations of benign uterine leiomyoma (fibroids) are rare. Benign metastasizing leiomyoma (BML) comprises an uncommon variant characterized by metastatic lung nodules. The pathologic characteristics for BML are well known in the literature; however, the underlying biology and molecular mechanisms remain poorly understood.We present a case of a 43-year-old woman who presented to the hospital complaining of dyspnea and lower extremity edema. Medical history includes a previous hysterectomy for leiomyomata two years prior. A reduced ejection fraction and right atrium globular filling defect are seen on transthoracic echo (TTE). CT scans of the chest, abdomen, and pelvis reported pelvic mass with an extensive inferior vena cava (IVC) thrombus extending into the right atrium, which was subsequently completely resected. Subsequent histopathology for the thrombus reported intravascular leiomyomatosis (IVL) and pelvic mass reported benign leiomyoma. Two years later, the symptoms recurred, and a chest CT revealed new pulmonary nodules. Subsequent pathology from a biopsy of these nodules was consistent with BML with ER+/PR+ on immunohistochemical staining.Genetic testing showed amplification of JUN, cyclin-dependent kinase 4 (CDK4), and MCL1, and loss of SUFU, AT-rich interaction domain 1A (AR1D1A), RB transcriptional corepressor 1 (RB1), and hepatocyte nuclear factor 1-alpha (HNF1A). The patient deemed to be a poor surgical candidate, and, therefore, she was started on hormonal treatment with leuprolide and letrozole. The disease remained stable upon follow-up at 48 months. Here, we report novel genomic profiling findings for the first time in a patient with a newly diagnosed BML. These findings may suggest molecular evidence that IVL may not be as benign as previously thought. Our study further highlights the value of genetic profiling in the understanding of this tumor's behavior and identification of new patient-specific therapeutic targets. Benign metastasizing leiomyoma (BML) is an uncommon condition with approximately 200 cases reported in the literature since the case first described by Steiner in 1939 . Our current understanding of this condition is limited to the cytogenetic level. Novel biomarkers have the potential to help risk-stratify patients with BML, thus enabling the development of a novel and precise molecular-guided therapeutic approach to management . Here weA 43-year-old Hispanic woman who had a past medical history relating to hypertension, obesity, and stroke was first admitted to the hospital in December 2014. There she had undergone hysterectomy for abnormal uterine bleeding, and surgical pathology, and at that time, was confirmed as having uterine leiomyoma. Two years after the hysterectomy, the patient was sent to the ED from the cardiology clinic for dyspnea, dizziness, and multiple episodes of syncope. Physical examination was within normal limits except for the presence of jugular venous distension, and irregular heart rate and rhythm where electrocardiogram (EKG) showed atrial fibrillation and transthoracic echo (TTE) reported nonischemic cardiomyopathy with a left ventricular ejection fraction (LVEF) of 20%-25% and a globular mass measuring 4.0 cm x 3.5 cm, almost filling the entire right atrium. Further workup, including abdominal ultrasound, revealed an enlarged inferior vena cava (IVC) with an intraluminal thrombus and occlusive portal vein thrombus causing absent flow consistent with Budd-Chiari syndrome. CT scan of the abdominal pelvis reported an extensive 5.7 cm x 4.7 cm IVC thrombus extending contiguously from the right mid external iliac vein and the left common iliac vein through the IVC and into the right atrium, in addition to a lobulated 12.0 cm pelvic mass Figure , B. MRI  A few months later, the patient also underwent an implantable cardioverter-defibrillator (ICD) placement for atrial fibrillation, and the patient's cardiac and functional status further improved, and she was able to get back to work. Two years later, the patient started complaining of an increasing lower extremity swelling, and dyspnea on exertion. A chest CT scan reported new pulmonary nodules Figure . Transjugular IVC mass biopsy performed, and pathology reported findings consistent with BML given the neoplasm exhibited staining with antibodies directed against S-100 and smooth muscle actin, without significant reactivity for antibodies directed against cytokeratins AE1/AE3 with ER+/PR+ on immunohistochemical stains. In light of her progressive disease, and worsening LVEF to 15%-20% upon preoperative evaluation, a multidisciplinary team recommended medical treatment consideration, and the patient was commenced on a new regimen of leuprolide and letrozole. While on this treatment, her abdominal mass and lung nodules were considered stable by RECIST 1.1 criteria , and she has had no new symptoms five years after initial diagnosis Figure . Molecular profilingMolecular profiling of the tumor showed amplification of cyclin-dependent kinase 4 (CDK4), JUN, and MCL1, and loss of AT-rich interaction domain 1A (AR1D1A), RB transcriptional corepressor 1 (RB1), SUFU, and hepatocyte nuclear factor 1-alpha (HNF1A)  is a common benign smooth muscle tumor in reproductive-age women. It has a spectrum of rare metastatic extrauterine variants, including IVL and BML. IVL characterized by benign smooth muscle tumor cells invade the uterine, pelvic veins, the vena cava, and may extend to the right heart. It is usually benign and susceptible to resection, but it can be fatal from resultant venous obstruction or cardiac involvement -8. An adOn the contrary to aggressive metastatic leiomyosarcoma, BML has been characterized by low mitotic counts, slow-growth, and thus, their size, number, and clinical symptoms usually remain stable unless they are diagnosed late because of symptoms from tumor size. Treatment of pulmonary BML depends on the patient’s age, disease extent, and the estrogen receptor (ER) status; it may include hysterectomy with oophorectomy, debulking surgery and/or the use of GnRH analogs, aromatase inhibitors, selective ER modulators, or chemotherapy to decrease tumor burden in conditions that are unamenable for surgery , 13-15. Our genomic analysis of the tumor sample identified multiple gene alterations Table  of thoseCarcinogenesis is a dynamic multistep process involving the progressive accumulation of genetic and epigenetic alterations that ultimately transform normal cells to neoplastic cells. Molecular alterations that drive malignant transformation and progression, often involve oncogenes and tumor suppressor genes.In this study, the next-generation sequencing findings of amplification of multiple oncogenes and loss of tumor suppressor genes suggest molecular evidence of malignant behavior of uterine leiomyomas. Further larger studies may help further advance our understanding of BML and better targeted therapeutic approaches. This report describes a case of BML, for which molecular profiling of a tumor biopsy revealed several novel genomic amplifications that have not been previously implicated in BML. These findings propose a novel molecular association with malignant expression in patients with uterine leiomyomas. Future research is needed to help improve our knowledge about this tumor's behavior and explore new patient-specific therapeutic targets.


Stoma-related complications are not rare, whereas the spontaneous perforation of the stoma limb is relatively rare. Herein, we report a case of stoma limb perforation which occurred after Hartmann’s operation.A 50-year-old Japanese man presented to our Hospital with acute and severe abdominal pain. Abdominal computed tomography (CT) scan revealed that an abscess with free air was formed around the sigmoid colon. We performed Hartmann’s operation, whereas he experienced redness, purulent discharge, and swelling around the colostomy at 10 days postoperatively. The contrast-enhanced CT scan of the abdomen revealed an abscess formation with air around the colostomy. He was diagnosed with an abdominal wall abscess due to perforation of the stoma limb.After the drainage, his symptoms were ameliorated by oral analgesics, anti-inflammatory drugs, and prophylactic antibiotic. Four months after the first operation, we performed a closedown of the sigmoid colostomy and fistula resection. The patient’s postoperative course was uneventful, and he was discharged 14 days later.This case depicts rare complications of Hartmann’s operation. Operation is usually performed in patients with stoma limb perforation. However, if they are stable and the abscess is located in their abdominal wall, they may be treated successfully using a multi-stage approach of local drainage toward the stoma wall followed by stoma closure. Stoma-related complications are with a frequency of 10–70%. Peristomal dermatitis, parastomal hernia, stomal prolapse, and subsidence are common, whereas the spontaneous perforation of the stoma limb is relatively rare . The proA 50-year-old Japanese man presented to the Department of the Gastroenterological Center at Shunjukai Shiroyama Hospital with acute and severe abdominal pain.An abdominal computed tomography (CT) scan revealed that an abscess with free air was formed around the sigmoid colon. Therefore, the patient was admitted to our hospital for operative treatment of the perforated sigmoid colon due to diverticulitis. Hartmann’s procedure was performed under general anesthesia with the patient in the supine position. The total operating time was 188 min, and the intraoperative blood loss was 200 ml.3, and the C-reactive protein (CRP) level was 10.12 mg/dl  laceration and thinning in the bowel wall caused when dehydrated fecaloma passes through the colon, (2) a disorder of circumstance with hyperextension bowel wall, and (3) inordinate pressure applied to the fragile bowel wall , 17. It Prognosis in colon perforation is generally unfavorable although improvements in diagnostic technology and treatment have resulted in reducing the mortality rates. However, there is no standard treatment for stoma limb perforation. Surgical treatment for abscess draining is performed regardless of a patient’s clinical, laboratory, and radiological findings. When emergency procedures are not warranted, a multi-stage or elective operative treatment algorithm may be more effective . This coWe diagnosed our case with spontaneous perforation of the colon because there was no macroscopic lesion, injury, and dehydrated fecaloma in the bowel and abdominal cavity. This is in line with the criteria of spontaneous perforation. Moreover, it is likely the stoma limb was weakened and perforated due to the poor condition of the colon wall coincident to increased bowel pressure. As the patient was in a stable condition and the abscess well-localized, we were able to perform the local drainage toward the wall around the stoma. The follow-up period after the local drainage was uneventful; therefore, we performed perforation excision and stoma closure instead of reconstruction of the colostomy.In conclusion, we presented a case with a rare complication of Hartmann’s operation. Patients with stoma limb perforation are usually treated operatively. However, if their general status is stable and the abscess is located in their abdominal wall, the patient may be treated successfully with a multi-stage approach of local drainage toward the wall followed by stoma closure.


Dermatofibrosarcoma protuberans is a rare mesenchymal tumor; it is locally aggressive and presents high rates of local recurrence. It may present as a nodular or plaque vegetating lesion. It mainly affects the trunk and proximal limbs, being rare in the distal extremities. Biopsy and immunohistochemistry help confirm the diagnosis. The authors report a case of dermatofibrosarcoma protuberans with plantar region involvement, a rare presentation. To the best of the authors’ knowledge, only 11 cases of involvement of the feet were described in the international literature. Dermatofibrosarcoma protuberans (DFSP) is a mesenchymal neoplasm of slow growth, locally aggressive and with low metastatic potential. It has several presentations and can be characterized as a hardened plaque, nodule, or vegetating lesion.Male patient, 35 years old, previously healthy, complained of painful lesion on the left plantar region with progressive growth over six months; he denied trauma. On physical examination, he presented an erythematous, exophytic, vegetating, and ulcerated tumor on the first metatarsal joint . HistopaDFSP is a rare mesenchymal tumor with low rates of aggressiveness. It presents slow progression, with high rates of local recurrence and rare cases of distant metastases. An American study conducted between 2000 and 2010 found an incidence of 41 cases in 10 million patients.A literature review published in 2019 described 11 cases of dermatofibrosarcoma that affected the feet. The mean age observed was 41 years; it was more frequent in men than in women (8:3) and the most frequent location of the tumor was the dorsum of the feet. The mean growth period was 3.5 years. The mean DFSP size at diagnosis was approximately 3.5 cm. Clinical diagnosis is difficult given the variety of presentations. Dermoscopy has been described as a useful tool to aid diagnosis.Histologically, dermatofibrosarcoma presents monomorphic spindle cells with little atypia and mitotic activity, arranged in irregular and multidirectional (storiform) fascicles. The tumor infiltrates the subcutaneous tissue, creating the characteristic honeycomb pattern. The histological differential diagnoses of other spindle cell tumors are dermatofibroma, malignant fibrous histiocytoma, atypical fibroxanthoma, desmoplastic melanoma, Kaposi's sarcoma, and solitary fibrous tumor.Immunohistochemistry becomes an important resource for differential diagnosis, as DFSP is positive for CD34 and negative for S 100 protein, factor XIIIA, and desmin.The recommended treatment is lesion excision with 2–3 cm of safety margins.Attention should be paid to this disease, even in unusual anatomical areas, as the prognosis depends on the early diagnosis and treatment.None declared.Simone Perazzoli: Conception and planning of the study; elaboration and writing of the manuscript; critical review of the literature.Renan Rangel Bonamigo: Approval of the final version of the manuscript; conception and planning of the study; elaboration and writing of the manuscript; obtaining, analyzing, and interpreting the data; effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases; critical review of the literature; critical review of the manuscript.Renata Heck: Approval of the final version of the manuscript; conception and planning of the study; elaboration and writing of the manuscript; obtaining, analyzing, and interpreting the data; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases.André da Silva Cartell: Approval of the final version of the manuscript; conception and planning of the study; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases.None declared.


Prototheca. In humans, protothecosis, caused mainly by P. wickerhamii, manifests itself in three forms: cutaneous, articular and systemic. It can occur in both immunocompetent and immunosuppressed individuals, being much more common in the latter. We present a new case of protothecosis in Brazil in a kidney transplant recipient.Protothecosis is a rare condition caused by the aclorophylated algae of the genus  Prototheca. Five species are known, two of which affect humans: P. zopfii and P. wickerhamii, the latter being the most common.Protothecosis is an uncommon infection caused by the aclorophyllated algae of the genus The first case of human infection was described in 1964 as a cutaneous ulcer located on the foot of a rice grower in Sierra Leone.A 60-year-old male patient, a skin phototype IV, born in Caetité-BA and moved to São Paulo-SP 35 years ago, reported a nodular lesion 6 months ago on the right leg , with noThe initial diagnostic hypothesis was subcutaneous mycosis and the patient was referred for complete lesion excision. The material was sent for histopathological analysis and granulomatous inflammatory process was observed with suppuration and sporangia grouped within the cytoplasm of the macrophages . The spoIn the postoperative period, the surgical wound was allowed to heal by secondary intention and was initially performed with compressive cotton dressing, then dressed daily with topical mupirocin for 3 weeks, and silver nitrate 10% until complete healing. The patient was treated with fluconazole 150 mg/day for a period of 3 months, until complete healing of the lesion.Prototheca do not present chloroplasts, organelles that contain the chlorophyll pigment, so they do not have the capacity to perform photosynthesis. They need a heterotrophic source of nutrients, such as organic carbon and nitrogen, and the skin is the most frequently affected organ.The algae of the genus Prototheca spp. is widely distributed throughout the world. Algae are found in soil, in the decomposition of plants, in water, and in certain food items. They also exist as a saprophyte organism in the human skin, nails and respiratory and digestive tracts.6Prototheca spp. resists chlorine treatment, sewage treatment and intestinal digestion. This contributes to its persistence in sewage, dissemination by domestic animals and permanence in the environment. It is also resistant to milk pasteurization, representing a problem for the consumption of dairy products.Human protothecosis is rare, but the incidence is higher in immunocompromised patients  than P. wickerhamii (3–15 μm).P. wickerhamii is to exhibit sporangiospores with a rounded central endospore, and this characteristic has been described as morula-like, daisy-like or raspberry-like.In histopathological examination, there is a granulomatous inflammatory infiltrate, consisting of lymphocytes, macrophages, giant cells and neutrophils. In staining with PAS or Grocott, we can observe sporangia internally containing sporangiospores, which may be inside macrophages or free in the exudate. Sporangia are surrounded by a capsule that can vary from 7 to 30 μm; Prototheca spp. due to the rarity of the disease. Amphotericin B appears to be effective in cases of disseminated infection.There is currently no definite treatment for In conclusion, nodular or nodule-cystic lesions in transplant recipients should be biopsied to screen for infectious agents using special stains . Culturing for fungi and mycobacteria is also recommended. The lesion of protothecosis should be completely excised and healing left by secondary intention. Complementing surgical treatment with azole derivatives is highly recommended.None declared.Valeria Romero Godofredo: Statistical analysis; approval of the final version of the manuscript; conception and planning of the study; elaboration and writing of the manuscript; obtaining, analysis, and interpretation of the data; effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.Milvia Maria Simões e Silva Enokihara: Intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the manuscript.Jane Tomimori: Obtaining, analysis, and interpretation of the data; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.Marilia Marufuji Ogawa: Approval of the final version of the manuscript; conception and planning of the study; effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.None declared.


Although spontaneous tumor rupture is a serious complication of hepatoblastoma, there is no consensus regarding the treatment strategy in infants. We report a patient with lung metastases who had a ruptured hepatoblastoma prior to the start of the scheduled chemotherapy and was successfully treated with a combined treatment including liver resection, lung resection, and chemotherapy.A 22-month-old boy with a ruptured hepatoblastoma and lung metastases underwent an emergency laparotomy with complete tumor resection, followed by chemotherapy. Moreover, a barely detectable metastatic lung lesion shown by a chest CT scan was resected after the fifth chemotherapy treatment. Both postoperative and chemotherapy courses were uneventful. The patient survived without any recurrent hepatoblastoma 2 years after the emergency surgery despite the poor prognosis indicated by distant metastases at the time of diagnosis.Because rupture itself can be the main cause of death in patients with hepatoblastoma, emergency tumor hemostasis is essential. However, there are no reports comparing the prognosis of the treatment method performing tumor hemostasis alone, tumor resection after chemotherapy, and tumor hemostasis and resection at the same time. The clinical course of the patient indicates that performing tumor hemostasis and resection simultaneously and lung resection after chemotherapy is an effective option to treat a ruptured hepatoblastoma with disseminated tumors and lung metastases if the patient’s condition is stable.Aggressive treatment with surgery and chemotherapy is an effective option for ruptured hepatoblastoma with disseminated tumors and lung metastases in infants. However, it is often diagnosed at advanced stages when resection is impossible or distant metastasis has already occurred. Recently, multidisciplinary protocols with both surgery and chemotherapy have been established for the treatment of hepatoblastoma [Spontaneous rupture of the tumor is a serious complication of hepatoblastoma, and various treatments such as liver resection, transcatheter arterial embolization (TAE), and chemotherapy have been attempted ,3. Howev2A 22-month-old boy with the chief complaint of an abdominal mass was referred to our hospital. An elastic, hard mass was palpable in his right abdomen on physical examination . UltrasoWe planned chemotherapy to treat the hepatoblastoma and lung metastases. However, the day before the scheduled start of chemotherapy, the patient became lethargic and tachycardic, with unstable vital signs that led to shock that was unresponsive to conservative measures. His hemoglobin level was 6.8 g/L. We performed an emergency laparotomy because the patient’s small size precluded TAE. During the surgery, the left margin of the tumor ruptured and the tumor contents leaked into the abdominal cavity . The patThe patient received six courses of chemotherapy (modified SIOPEL-4 regimen) in the pediatric department . Chest C3In recent years, surgical resection with pre- and/or post-operative chemotherapy has markedly improved the survival rate of patients with hepatoblastoma . However4We are focusing on the usefulness of temporary tumor resection at the time of rupture as well as the removal of tumor contents that have disseminated intraperitoneally while still being confined. This type of procedure can lead to a good clinical course in young patients. Even if the vital signs deteriorate temporarily at the time of tumor rupture, it may be possible to resect the tumor temporarily if vital signs stabilize after the start of conservative treatment.Although there is no consensus on the treatment of ruptured hepatoblastoma in infants, we believe it is important to actively treat hepatoblastoma cases that include distant metastases.All authors declare that they have no conflicts of interest.All authors declare that they have no funding source.This submission is a case report about surgical strategy, not the manuscript reporting studies involving human participants, human data or human tissue.In this case, ethical approval has been exempted by our institution.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Masaki Horiike (MH) made the conception and design of this case report. Authors other than MH contributed to the collection, analysis, and interpretation of the data. MH wrote the draft manuscript, and other authors performed the critical revision of the manuscript. All authors gave final approval of the version to be published. MH has overall responsibility and guarantees the scientific integrity.1.Name of the registry: Masaki Horiike.2.Unique identifying number or registration ID: Researchregistry5920.3.https://www.wakayama-med.jrc.or.jp/department/shonigeka/.Hyperlink to your specific registration (must be publicly accessible and will be checked): MH has overall responsibility and guarantees the scientific integrity.Not commissioned, externally peer-reviewed.


Ganglion cell-containing pituitary adenomas that are neurofilament protein-positive are the exceedingly rare form of pituitary ganliocytomas. We report a case of a 23-year-old male patient who presented with a clinical picture of acromegaly in addition to raised prolactin level. Histopathology showed areas exhibiting pseudopapillary and solid proliferation of round and monotonous blue cells. The immunohistochemistry showed strong reactivity for synaptophysin and growth hormone and focal reactivity to prolactin. Fibrous bodies are confirmed using cytokeratin immunostain, in keeping with sparsely granulated somatotroph component. The patient remained free of recurrence after one year of radiological follow-up. Pituitary macroadenomas are benign pituitary gland tumors with an estimated 10-25% overall prevalence among all intracranial neoplasms that grow to at least 1 cm in size . PituitaA 23-year-old male patient presented with a clinical picture of acromegaly. GH level was 40.33 ng/mL (reference < 10 ng/mL), insulin-like growth factor 1 (IGF-1) was 1056 ng/mL (reference 96-228 ng/mL), and prolactin was 759 mIU/L (reference 86-324 mIU/L). Magnetic resonance imaging showed that the sella turcica was expanded by a pituitary macroadenoma measuring 2.1 × 1.8 × 1.9 cm . Mild enThe tumor was resected using a transnasal approach. Histopathological examination of the resected tumor samples showed two patterns . One wasGanglion cell-containing pituitary adenomas are very rare and usually show neurofilament protein- (NFP-) positive, dot-like areas of cytoplasmic reactivity that indicates neuronal differentiation within adenoma cells –7. TheseHistopathology of the resected tumor in our patient showed round blue cell areas that look atypical on low power. These areas are located at the edges of the neuropil-rich area. Round cells are typically noted in sparsely granulated somatotroph pituitary adenoma. The tumor cells in this case also showed paranuclear inclusion (fibrous bodies) on Cytokeratin stain, which is a feature of sparsely granulated somatotroph. No mitosis or necrosis was seen, and the Ki-67 proliferative index was low. This may indicate a lower probability of postsurgical progression. Some studies have shown that immunohistochemistry for Ki-67 of pituitary adenomas with a proliferation index greater than 1.3% predicts the progression of the disease . HoweverAn interesting feature in this case is the prolactin reactivity, which was highlighting neurons and their sprouting axons. Hence, the increased serum prolactin level was probably due to the prolactin secretion of the neuronal compartment of the tumor.As per the WHO classification, this is a case of mixed gangliocytoma-pituitary adenoma. Different theories for the origin of pituitary adenomas with neuronal elements are suggested. Thodou et al.  summarizIn our case, the tumor showed prolactin stain highlighting small cells that exhibit attenuated neuronal axons. In a case series by Lopes et al. , the autLong-term monitoring of these patients may be necessary although most pituitary adenomas are generally slow-growing with low proliferative index. It was suggested that if no evidence of tumor progression is shown after 3-5 years following surgery, monitoring of the patient should be extended for an additional period .


Endobronchial lipoma is an extremely rare benign tumor, which is generally located in the first three subdivisions of the tracheobronchial tree. According to the existing literature, all endobronchial lipomas are single (one per patient). Here, we report a rare case in which the patient presented with two endobronchial lipomas in the same patient, and underwent a bronchoscopic tumor resection in the left main bronchus and the left lower bronchus. Both tumors were pathologically confirmed as endobronchial lipoma.A 52-year-old Chinese man presented at the clinic reporting a mild cough with yellow color sputum and exertional dyspnea for 2 weeks. He was a heavy smoker (45 pack-years). Chest auscultation demonstrated faint wheezing in left lower lobe. Computed tomography (CT) revealed two low-density endobronchial masses located in the middle segment of the left main bronchus and the posterior basilar segmental bronchus of the left lower lobe. The neoplasms measured a CT-attenuation value of -70HU, −98HU in density with air trapping and atelectasis in the segmental bronchus of the left lower lobe. The patient underwent interventional bronchoscopic management to remove the neoplasms by using an electrosurgical snare, cryotherapy, and electrocautery. The locations of the neoplasms were confirmed at the left main bronchus and the superior segment of the left lower lobe during bronchoscopic intervention. Histopathological examination confirmed that both tissues were consistent with lipomas. After 18 months of follow-up, the patient was free of symptoms and CT revealed that bronchiectasia remained in the superior segment of the left lower lobe; however, no mass lesion was present in the left bronchus.This case suggests that an endobronchial lipoma can present as multiple lesions, and both proximal and distal types can simultaneously occur in the same patient. Thus, these findings help us further understand the biology of endobronchial lipomas. Endobronchial lipoma is an extremely rare benign tumor, comprising approximately 0.1–0.5% of all bronchial tumors . When th2 was 98% when breathing ambient air, the respiratory rate was 20/min, and the blood pressure was 119/77 mmHg. Chest auscultation demonstrated faint wheezing in left lower lobe. A computed tomography (CT) scan was performed, which revealed two low-density endobronchial masses located in the middle segment of the left main bronchus and the posterior basilar segmental bronchus of the left lower lobe. The neoplasms measured the CT-attenuation value -70HU, −98HU  and had a history of a splenectomy for abdominal injury 23 years ago. After a failed 3 day-course of antibiotic treatment at the clinic, he was referred to our hospital for further examination and treatment. Upon physical examination, the body mass index (BMI) was 17.97, SpOIn view of the above, interventional bronchoscopic management was undertaken to remove the neoplasms. First, we therapeutically resected the neoplasm, of which the base was located in the lateral wall of the left main bronchus using electrosurgical snare under flexible bronchoscope Fig. . Then, tAfter bronchoscopic intervention, the patient recovered and was discharged. After 18 months of follow-up, the patient was free of symptoms and CT revealed that bronchiectasia remained in the superior segment of the left lower lobe Figs.  and 12, Endobronchial lipoma is a type of rare benign tumor, which originates from fat cells located in peribronchial and submucosal tissues of the bronchi . AlthougFor most cases, lipomatous neoplasms arise in the setting of lipomatosis and rarely involve the tracheobronchial tree , 5. EndoEndobronchial lipomas grow slowly and have different clinical manifestations . The cliAt present, a chest CT and bronchoscopy are main approaches to diagnose endobronchial lipoma as the sensitivity of traditional X-ray examination is lower (66%) , 15. TheEndoscopic features of tracheobronchial lipomas have been classified as follows. These tumors always appear with a smooth surface and are oval-shaped, only a few could be lobulated, and they are poorly vascularized with a yellow to rose appearance. Furthermore, the mobility of the tumors is in general mobile, it is rarely to see fixed one. Finally, the proportions of proximal and distal types are similar when it comes to the location of the tumors , 12. TheThe treatment of endobronchial lipomas includes two methods: bronchoscopic resection and surgery . With thIn summary, in this case we reported multiple endobronchial lipomas that coexisted proximal and distal, indicating a novel discovery of endobronchial lipoma, as all cases previously reported were single. We demonstrated that the clinical features, radiological features, endoscopic features, and histopathological features supported previous observations. Taken together, this case helps us further understand the biology of endobronchial lipomas.


Soft-tissue sarcomas (STS) are rare in hand and foot. In this paper we present a case of reconstruction of Achilles tendon defect with peroneus brevis transfer reinforced with medial gastrocnemius fascia and plantaris tendon after excision of a local recurrence of epithelioid sarcoma.Fifty-five years-old female. MRI showed a lump of 5 × 2,5 × 2 cm into Achille’s tendon with invasion of the anterior fat tissue but no invasion of the surrounding bones. The patient underwent excision of the tumour and reconstruction of the tendinous defect with peroneus brevis transfer. Surgical technique has been widely described.Epithelioid sarcoma arising from the Achilles tendon is an extremely rare malignant tumour in an atypical site and may easily be confused with other soft tissue masses. It presents a technical challenge because of the large tendon defect remaining following wide resection. Reconstruction with peroneus brevis transfer, reinforced with medial gastrocnemius fascia and plantaris tendon, restore appropriate structural continuity and resistance. Functional results are satisfactory. Soft-tissue sarcomas (STS) are rare malignant mesenchymal tumours which arise in head, neck, retroperitoneum and limbs in most of the cases. They are rare in hand and foot . The mosGenerally, ES grows in subcutaneous layers, presenting as firm solitary or multinodular masses which may ulcerate and sometimes can be misdiagnosed as ulcerating squamous carcinoma. Deep-seated tumours are typically larger and firmly attached to tendons and fascia. Soft tissue sarcomas, arising from tendons and aponeuroses are rare and only a few cases are described in the literature , 8.Because of its uncharacteristic clinical features, ES is easy to misdiagnose. Correct diagnosis is crucial not to income to inadequate excision , 7.Soft tissue sarcomas of the foot represent a specific reconstructive challenge to the orthopaedic surgeon because of their proximity to critical structures such as tendons, ligaments, bones and neurovascular structure. In order to obtain wide margins, sacrifice of some of these structures is often necessary, with important functional impairment . In addiIn this paper we present a technical case of reconstruction of Achilles tendon defect with peroneus brevis transfer reinforced with medial gastrocnemius fascia and plantaris tendon after excision of a rare case of local recurrence of epithelioid sarcoma as an effective reconstructive strategy to obtain satisfactory functional results even after a wide resection as required.A 55 years-old female presented with insidious right foot pain and swelling over 6 months in absence of trauma. Symptoms worsened with walking and standing.In April 2007 the patient underwent unplanned excision of the lump in the right Achilles’ tendon. Histology showed a myxofibrous inflammatory sarcoma grade 2 according to FNLCC classification.Magnetic resonance imaging (MRI) was negative for local recurrence at 6 and 12 months after surgery. No further follow-up was performed during the next 4 years.In August 2012 the patient was referred to our Institute for pain lasting 2 years. At the physical examination, the foot presented a lump, slightly painful, attached to the Achille’s tendon, with no signs of infection.In October 2014 MRI showed a lump of 5 × 2,5 × 2 cm into Achille’s tendon with invasion of the anterior fat tissue but no invasion of the surrounding bones Fig. . SurgicaForty days later the patient was re-admitted for plaster cast removal and to start adequate physiotherapic protocol. She was allowed to walk with progressive weight bearing, wearing ankle brace.Follow up visits, Thoracic computerized tomography (CT) and foot and ankle MRI were then scheduled at every 4 months for 3 years, then every 6 months until the fifth year after surgery and then annually until the tenth year.In January 2015 the patient presented a small area of skin defect and in February she was treated with negative pressure wound therapy for 3 months. In June the skin was completely healed, the MRI showed no sign of local recurrence Fig.  and the One year later ankle range of movement was 45° in flexion, with a good ankle stability.At the final follow-up at 5 years in 2019 there was no evidence of oncological disease and the patient was able to walk without pain nor limping, she could walk on tiptoe and remained a slight defect of 10° of flexion comparing to the contralateral side.Pre-operatively, the insertion of the Achilles tendon on the calcaneum and the insertion of the peroneus brevis tendon over the base of the fifth metatarsal were identified as landmarks.Under spinal anaesthesia, the patient was placed in prone position, with feet dangling from the end of the operating table and a thigh tourniquet was applied and inflated.A 10 to 12 cm longitudinal skin incision was made directly on the median axis of the Achilles tendon; the previous incision was excised and lengthened for adequate exposure. Subcutaneous fat was penetrated by means of sharp dissection, sural nerve isolated and protected. The Achilles tendon was thus exposed, gently isolated along its length and visually examined. The tendon itself was adhered to surrounding paratenon and subcutaneous tissue and it appeared thickened and of reduced consistency. Ventrally, the loose tissues appeared undamaged and clean from gross traces of neoplasia, except for a small tumour expansion toward the anterior fat tissue.We proceeded to separate the lesion from surrounding tissues and to resect en bloc a 6 cm segment of tendon, from the calcaneal apex to the proximal clean margin. Remaining tissue appeared healthy and skin edges viable.Through the floor of the wound, the deep fascia overlying the compartment containing the peroneal muscles could be seen. The deep fascia overlying the peroneal tendons was incised and the peroneal tendons were mobilized. The distal portion of peroneus brevis muscle belly was exposed and its proximal end was identified.A 2.5 cm longitudinal skin incision was made over the base of the fifth metatarsal and the tendon was identified and exposed. A stay suture was applied in the distal end of the tendon and this was detached from its insertion. After smooth dissection of the sheath, the tendon was delivered, by gentle traction, through the subcutaneous tissue, pulled through inferior retinaculum and basted in the distal end. The peroneus brevis tendon was then passed through a trans-osseous tunnel made through the posterior calcaneal tuberosity from medial-to-lateral and pulled proximally (Fig. To ensure better function and resistance, the medial part of the gastrocnemius fascia was detached and distally overturned to support the structure. Following, the plantaris tendon was harvested, weaved through the Achilles stump and sutured laterally to peroneus brevis tendon to augment the reconstruction. Tourniquet was deflated and hemostasis done. Subcutaneous tissue and the skin were closed, being careful of tension. The wound was dressed, a below-the-knee plaster cast applied with the foot in gravity equinus.. Metastases from epithelioid sarcoma have been described in about 45% of patients with involvement of lungs and lymph nodes [STS are uncommon, they represent only 1% of all adult malignancies and less than 10% are located in the distal limb , 18. Epiph nodes , 16. TheThe goals of malignant tumor resection in the foot and ankle are to obtain a wide surgical margin and to maintain a sensate, plantigrade and functional foot. Because of its rarity and its uncharacteristic clinical features, ES is easy to misdiagnose. Correct diagnosis is crucial not to income into inadequate excision and consequent need for revision surgery. According to Koulaxouzidis et al.  the so cIn Literature several surgical reconstruction techniques have been documented, more often used after a traumatic or chronic rupture. Maffulli et al. , 11 descEpithelioid sarcoma arising from the Achilles tendon is an extremely rare malignant tumour in an atypical site and may easily be confused with other soft tissue masses. It presents a technical challenge because of the large tendon defect remaining following wide resection. Reconstruction with peroneus brevis transfer, reinforced with medial gastrocnemius fascia and plantaris tendon, restore appropriate structural continuity and resistance. Functional results are satisfactory.


Kaposi sarcoma (KS) is a vascular neoplasm caused by human gammaherpesvirus 8 (HHV-8). Four subtypes of KS are described: classic (Mediterranean), epidemic (acquired immunodeficiency syndrome (AIDS)-associated), endemic (sub-Saharan Africa), and iatrogenic. Iatrogenic KS due to tumor necrosis factor-alpha (TNF-α) inhibitor therapy is particularly rare. A 66-year-old female with a history of seropositive rheumatoid arthritis (RA) presented with a skin lesion on her right second toe. Diagnosed with RA four years prior, she failed to respond to methotrexate, hydroxychloroquine, and etanercept. As a result, she was started on adalimumab. Approximately two months into therapy, she presented to the emergency room with a dark brown skin lesion on her right second toe. She underwent excisional biopsy of the mass, which demonstrated a tumor composed of spindle cells forming slit-like spaces with extravasated red blood cells. The tumor was positive for cluster of differentiation 31 (CD31), CD34, and HHV-8 immunostains and negative for smooth muscle antibody (SMA) and desmin immunostains, consistent with Kaposi sarcoma. Human immunodeficiency virus (HIV) serology was negative. The patient was diagnosed with iatrogenic KS. Adalimumab was discontinued. The patient was started on alitretinoin and underwent adjuvant radiation therapy to minimize recurrence. TNF-α is a pro-inflammatory cytokine that has been implicated in many inflammatory diseases and in cell apoptosis. While anti-TNF-α agents have improved outcomes in many immune-mediated diseases, higher rates of infections and malignancy have also been reported. The incidence of KS with anti-TNF-α therapy remains a rare entity. Therefore, it is extremely important for patients receiving biologic agents, including TNF-α inhibitors, to have a close follow-up and receive routine skin evaluation for malignancy. Clinicians should have a high index of suspicion for KS in such non-HIV patients started on immunosuppressive agents. Kaposi sarcoma (KS) is a vascular neoplasm caused by Kaposi sarcoma herpesvirus (KSHV), also known as human gammaherpesvirus 8 (HHV-8) . Kaposi A 66-year-old female of middle eastern ethnicity with a history of seropositive rheumatoid arthritis (RA) was noted to have a skin lesion on her right second toe. Diagnosed with RA four years prior, she had failed to respond to methotrexate, hydroxychloroquine, and etanercept. As a result, she was started on adalimumab 40 mg every two weeks. Approximately two months into adalimumab therapy, the patient presented to the emergency room with a dark brown skin lesion on her right second toe. The lesion had been present for approximately three to four weeks, was slowly increasing in size, with intermittent tenderness and occasional bleeding. She did not report a history of diabetes or recent foot injuries. Physical exam showed a raised, brown lesion over the distal, dorsolateral second digit, approximately 1.4 cm in size, well-circumscribed from the surrounding tissue of the digit. A magnetic resonance imaging (MRI) scan revealed a 15.95 mm x 10.67 mm x 9.25 mm oval, circumscribed, exophytic, T1 intermediate intensity, T2 intermediate intensity mass in the subcutaneous soft tissues of the lateral second digit without the involvement of the underlying second extensor tendon or the second middle phalanx Figures -1B.She subsequently underwent an excisional biopsy, which microscopically demonstrated a tumor composed of spindle cells forming slit-like spaces with extravasated red blood cells Figures -3. The tHuman immunodeficiency virus (HIV) serology was negative. Adalimumab was discontinued. The patient was started on alitretinoin and underwent adjuvant radiation therapy to minimize recurrence.TNF-α is a pro-inflammatory cytokine that has been implicated in numerous inflammatory diseases and in cell apoptosis . TreatmeTo our knowledge, five cases of KS were identified with the use of infliximab, two cases with adalimumab, one with golimumab, and one with certolizumab pegol -15. In mRare reports of iatrogenic Kaposi sarcoma in non-HIV patients receiving anti-TNF-α therapy have been identified. Patients receiving TNF-α inhibitors should undergo routine skin evaluation for malignancy.


In front of a colonic tumor, the diagnosis of a pseudotumoral form of Crohn's disease must be considered. However, it is a rare form, especially when inaugural and a neoplasia must be eliminated before retaining the diagnosis. In front of a colonic tumor, the diagnosis of a pseudotumoral form of Crohn's disease must be considered. However, it is a rare form, especially when inaugural and a neoplasia must be eliminated before retaining the diagnosis. A neoplasia must be eliminated in front of a colonic tumor before retaining the diagnosis, hence the need of macrobiopsies or even pathological evidence on the operating specimen. A case of a 44‐year‐old patient in whom the diagnosis of inaugural Crohn's disease in its pseudotumoral form was retained on the data of the pathological examination of the operating specimen after a right hemicolectomy is reported. A review of cases presented in the literature is also performed.2A 44‐year‐old male patient, with history of appendectomy in 1994, operated twice for perianal abscesses in 2017 and 2018, consulted the emergency department for abdominal pain and subocclusive syndrome associated with deterioration in general condition with a weight loss of 10 kg.3, CRP = 11.2 mg/dL), a hypochromic microcytic anemia at 11.8 g/dL, and hypoalbuminemia at 28 g/L. The tumor markers (carcinoembryonic antigen) were normal.Abdominal examination found a painful 4 cm soft mass in the right iliac fossa and biological findings revealed a biological inflammatory syndrome  is indicated because of the presence of a risk factor for postoperative recurrence (tobacco) as recommended by the European Crohn's Colitis Organization3We report a case of Crohn's disease in its pseudotumoral form which remains an exceptional presentation of the disease. The main differential diagnosis in front of a colonic tumor is neoplasia which we must strive to eliminate, especially since Crohn's disease can degenerate into adenocarcinoma. Meanwhile, other differential diagnosis should also be considered as intestinal tuberculosisIn fact, this pseudotumoral form of Crohn's disease has been already reported in the literature. Its clinical presentation is variable: abdominal mass like our case and the case of Stoica et al,The abdomino‐pelvic CT scans performed in most of these patients with a colonic tumor were not contributory to the diagnosis of these pseudotumoral forms as they fail to differentiate them from colonic neoplasia. Thus, the diagnosis must be made by pathological examination.In fact, histological examination of macrobiopsies of the process helped the diagnosis of Crohn's disease in only three cases of Maamouri et al,In our patient case, we tried to obtain a histological confirmation to avoid surgery, as medical treatment can be enough in Crohn's disease. We performed biopsies of the colonic process twice but they were not contributory to the diagnosis. Hence, the patient was operated on to eliminate a neoplasia and have the definitive diagnosis.Two of the patients in whom the diagnosis was made on macrobiopsies, clinical,The cases reported in the literature are summarized in Table 4The pseudotumoral form of Crohn's disease is exceptional and appears to have its own clinical, morphological, and progressive features. The clinical and the endoscopic presentations are variable and the course of treatment is often favorable. It is probably not very aggressive compared to other forms of Crohn's disease. However, its characteristics remain to be defined.None declared.MS and GG: wrote the paper. GG: reviews the literature. RJ: contributes by the pathology pictures as well as the interpretation of figures. DH: operated the patient on. NB: referring Doctor. CC: contributes by the CT scan pictures. DG: The head of the gastroenterology department in the Hbib Thameu Hospital and contributes in the therapeutic decisions.Patient personal data have been respected.


Angiosarcoma occurs very rarely in the oral cavity, and the epithelioid type is even rarer. Here, we report a rare case involving an elderly man with a primary epithelioid angiosarcoma that originated from the mandibular gingiva and resembled a dentigerous cyst on radiographs.A 66-year-old Japanese man visited our hospital with a chief complaint of gingival swelling in right mandibular third molar region. A panoramic radiograph showed bone resorption around the crown of right mandibular third molar, which was impacted. Incisional biopsy confirmed a diagnosis of epithelioid angiosarcoma. The lesion exhibited aggressive proliferation after biopsy resulting in uncontrolled bleeding and difficulty in closing the mouth. Mandibular segmental resection including the tumor was performed without reconstruction. Because of the aggressive preoperative course of the tumor, the patient received adjuvant chemotherapy. There were no signs of recurrence during a 2-year follow-up period.A review of the literature yielded only four reported cases of epithelioid angiosarcoma in the jaw region, with the lesions occurring in the maxilla in three cases. To our knowledge, this is the second case of primary epithelioid angiosarcoma in the mandible. Angiosarcomas account for only 2% of all soft tissue sarcomas –3. More Epithelioid vascular neoplasm is a unique tumor characterized by proliferating “epithelioid” or “histiocytoid” endothelial cells showing a sheet-like growth pattern with vascular differentiation –11. EpitUntil now, only four cases of primary EA in the jaw region have been reported, with three lesions originating in the maxilla and one in the mandible , 13, 14.A 66-year-old Japanese man visited a private dental office with a chief complaint of gingival swelling around right mandibular third molar. Considering the diagnosis of pericoronitis, the dentist performed incisional drainage and prescribed antibiotics and analgesics; however, the swelling reappeared with mild gingival bleeding 20 days after his first visit to the dental office. The dentist repeated incisional drainage and prescribed antibiotics, both of which were ineffective. The gingival swelling and oozing persisted; therefore, curettage of the swollen gingiva was performed. The dentist further referred the patient to our university hospital for extraction of the third molar, and he presented at our department 18 days after curettage of gingiva.The patient’s medical history included controlled hypertension and cholecystectomy for gallstones. Hematological and biochemical examinations did not reveal any abnormalities. His facial profile was symmetrical, and the submandibular lymph nodes showed no tenderness or swelling on ultrasound. Intraoral examination revealed a dark purple gingival swelling with easy bleeding around the crown of right mandibular third molar Fig. a. The maSurgical treatment with vascularized free flap reconstruction was planned. However, 4 weeks after the biopsy, the lesion exhibited aggressive proliferation perhaps because of traumatic irritation by the antagonist teeth. This led to uncontrolled bleeding and difficulty in closing the mouth Fig. c. The tr2) was administered once a week for 3 weeks, followed by a 1-week rest period. This cycle was repeated for 1 year. The patient showed no signs of recurrence during the 1-year follow-up period.The patient’s postoperative course was uneventful except for a delayed wound healing of the oral mucosa. Nevertheless, because the tumor had exhibited an aggressive preoperative course, he received adjuvant chemotherapy 2 months after the surgery . RadiatiHistopathologically, the tumor was composed of solid sheets with incomplete alveolar structures Fig. a and no EA is a rare endothelial cell malignancy. Epithelioid endothelial cell tumors include epithelioid hemangioma, epithelioid hemangioendothelioma (EHE), and EA . EpithelAngiosarcoma may be difficult to diagnose because of its various clinical and pathological characteristics . DiffereIn immunohistochemistry, positive staining for factor VIII, CD34, and CD31, which are endothelial markers, indicates that the tumor has endothelial characteristics. The lesion in our patient showed positivity for factor VIII, CD31, and vimentin. In particular, CD31, a membrane glycoprotein that is important for endothelial cell-cell interactions and vascular adhesion of leukocytes, is considered the best marker for the endothelial phenotype, especially the poorly differentiated variant . Our patMost authors believe that surgery in combination with radiotherapy and/or chemotherapy offers the best chance of survival . AngiosaWe reported a rare case of a primary EA originating from the mandibular gingiva in an elderly man. To our knowledge, this is the first report of EA in the mandible, and the findings highlight the importance of early diagnosis and adequate surgical resection, which should be performed as soon as possible.


To the Editor:1 Most of them are nonfunctional tumors (50%–75%).2 Newly published 2010 World Health Organization (WHO) classification classified them into three distinct types: neuroendocrine tumor grade 1 (G1), neuroendocrine tumor grade 2 (G2), and neuroendocrine carcinoma (G3).3 Pancreatic neuroendocrine carcinoma (pNEC) only amounts to about 2% to 3% of all pNETs. Here we demonstrated a rare case of pNEC with isolated retroperitoneal metastasis. It served as the only reported case of pNEC metastases spread to the retroperitoneal preceded to other organs.Pancreatic neuroendocrine tumors (pNETs) are rare neuroendocrine neoplasms, with an estimated incidence of approximately 1 per million per year, about 3% of primary pancreatic tumors.A 61-year-old female was referred to our hospital with gallbladder polyps and right upper quadrant pain. We discovered a 3.55-cm mass in the tail of the pancreas, a 3.92-cm mass in the right retroperitoneal, and right retroperitoneal lymph node enlargement  and fluorine-18-octreotide (18F-Octreotide) positron emission tomography (PET)/CT showed abnormal uptake of 18F-Octreotide was observed in newly found lesions and retroperitoneal metastasis recurrent, which were considered as NEC metastasis tumors  in oncology. Unfortunately, the effect of medical treatment was minimal and the patient ultimately succumbed 25 months after the surgery.After 20 months of regular follow-up, some liver, bone, and right kidney lesions with retroperitoneal metastasis recurrence were found by abdominal computed tomography (CT). Further fluorine-18-fluorodeoxyglucose Ga-labeled somatostatin analogs PET/CT have higher sensitivity (92%), higher specificity (88%), higher accuracy (93%), and empowers whole-body assessment of disease extent.6 Histopathological classification of pNETs can be categorized based on the mitotic count and the Ki-67 proliferation index according to 2010 WHO classification of gastropancreatic-neuroendocrine neoplasms. On this measure, the high of two is adopted for categorization, our case can be classified as G3. Some authors speculate that NEC with a Ki-67 index below 55%, can be called a new category, had better survival and lower relative risk.7 Many scholars hold the view that NEC may not have only 1 entity. By analyzing 2158 cases from the Surveillance, Epidemiology, and End Results database, grading and systemic metastases are considered to have a significant impact on survival.8The microscopic analysis combined with immunohistochemistry is the “gold standard” for the diagnosis of pNETs. Chromogranin A (CgA) and synaptophysin (Syn) are neuroendocrine markers.9 In contrast, for poorly differentiated (high-grade) extrapulmonary NECs, the North American Neuroendocrine Tumor Society guideline believes that it is unlikely to be curative by surgery alone. Chemotherapy (4 to 6 cycles of carboplatin and etoposide or cisplatin) after surgery is recommended.10Surgical resection with regional lymph node dissection is the only cure for patients with pancreatic NETs, but the role of chemotherapy after curative resection is still unclear. The National Comprehensive Cancer Network guideline recommends if complete resection is possible, resect the primary tumor and metastases with a regular postoperative review is enough.In summary, owing to the nonfunction, most of the pNETs can only be incidentally discovered by imaging. Surgical resection remains the only cure. Positron emission tomography/CT is one of best imaging techniques for whole body assessment of disease extent and valuable surgical or medical treatment. Chemotherapy after curative resection of pNEC may influence their prognosis. Medical management for advanced pNETs is of equal importance.Haoxiang Zhang, MDDong Shang, MD, PhDDepartment of General SurgeryThe First Affiliated Hospitalof Dalian Medical UniversityDalian, Chinashangdong@dmu.edu.cn


Hypothenar hammer syndrome (HHS) is a vascular disorder characterized by ulnar artery thrombosis or aneurysmal formation. It is most commonly reported in middle-aged males who suffer from repetitive trauma to the palm secondary to occupational or recreational activities. Most cases of HHS can be managed conservatively; however, surgical management is typically indicated for persistent symptoms following conservative measures, imminent vascular compromise, those who fail conservative treatment, or those in imminent danger of rupture. Here we present the case of a right-handed 74-year-old female with HHS who presented with a three-week history of a mass in the hypothenar eminence of the left hand. Reportedly, the mass had appeared slowly and had no associated pain, ischemia of the distal hand, numbness, paresthesia, or changes in the overlying skin. She denied any history of a blunt or penetrating hand injury. The patient was treated surgically by removing a 2.0-cm thrombosed pseudoaneurysm of a collateral branch of the ulnar artery within the left hypothenar eminence. On follow-ups at 1, 2, and 12 weeks postoperatively, the patient's pain was found to be well-controlled. Her normal range of motion was restored, and her digits remained neurovascularly intact. This was an atypical presentation of HHS, and our review of the disorder emphasizes the importance of diagnostic reasoning in rare conditions with unusual presentations of HHS. Hypothenar hammer syndrome (HHS) is a vascular disorder that predominantly affects the dominant hand of middle-aged men who utilize their hand as a “hammer”, arising from repetitive blunt trauma to the hypothenar eminence or, rarely, from a single traumatic blunt injury to the hypothenar eminence -5. This We present the case of a right-handed, 74-year-old female who presented with a three-week history of a mass in the hypothenar eminence of the left hand. According to the patient, the mass had appeared slowly and had no associated pain, ischemia of the distal hand, numbness, paresthesia, or changes in the overlying skin. She denied any history of a blunt or penetrating hand injury. The patient was a lifelong nonsmoker and had a past medical history significant for right breast cancer (status: post lumpectomy and radiation), with postoperative right upper extremity lymphedema.A physical exam of the left upper extremity showed a non-tender, non-pulsatile, firm 1.5 x 1.5-cm subcutaneous mass within the hypothenar eminence at the hook of the hamate  in 1,300 patients referred to their center for hand ischemia . A large.7% in 1,DiagnosisDiagnosis of HHS requires clinical suspicion and can be confirmed via different imaging modalities, such as the characteristic “ying-yang sign” on ultrasound or CT angiography ,6. On phCatheter-based angiography remains the gold standard to identify the lesion and nearby anatomy, especially if surgical intervention is being considered . CT angiTreatmentDue to the relatively rare nature of HHS, there is no consensus on the best management of the condition ,3,7. ConHHS is a relatively rare syndrome, typically presenting in middle-aged males who undergo repetitive trauma to the ulnar side of the palm. This case represented a unique presentation of HHS in a 74-year-old female with no history of trauma or repetitive activity to the hypothenar eminence. Also novel to this presentation was the occurrence of a thrombosed pseudoaneurysm in the non-dominant hand and the lack of associated symptoms, leading us to ponder on the etiology of this patient’s condition given the lack of any typical causative factors. This case, in particular, emphasizes the importance of diagnostic reasoning in rare presentations of HSS. Due to the unique presentation seen in the case above, the diagnosis could have been missed at the expense of morbidity to the patient.


The cause of peritonitis might be missed in conventional imaging.•Peritoneal toileting is more efficiently done by laparoscopy.•Diagnostic laparoscopy may avoid the hazards of unnecessary laparotomy. Acute peritonitis is a surgical emergency and is conventionally managed with exploratory laparotomy in many centers, but laparoscopy is not contraindicated in such conditions. Operative management remains a cornerstone treatment in peritonitis irrespective of the primary pathology.A 32-years-old man was admitted with acute diffuse peritonitis. As he was clinically stable, we proceeded with conservative treatment and diagnostic workup. He did not respond by 24 h and CT imaging showed ascites, but no definite diagnosis could be made. Diagnostic laparoscopy was performed, thorough peritoneal toileting was done but surprisingly the etiology was unclear. Following laparoscopy, he responded well with an uneventful recovery.Timely proper evaluation and correct intervention are a paramount concern for the positive outcome. Regarding the most effective operative approach (laparotomy or laparoscopy) in acute peritonitis remains controversial in the literature and laparoscopy has been considered as a relative contraindication. However, the laparoscopy is gaining wider acceptance as the limitations of laparoscopy are declining rapidly. We experienced a case of extensive peritonitis with unclear etiology which we managed successfully by laparoscopy.Laparoscopic approach in peritonitis is effective and helpful for accurate diagnosis and to minimize the hazards of an unnecessary laparotomy. This case report might encourage many surgeons to consider laparoscopic approach in peritonitis with confidence and to perform further studies. Peritonitis may be classified as primary spontaneous peritonitis and secondary peritonitis. Primary spontaneous peritonitis includes the risk factors like cirrhotic liver disease with ascites and renal failure needs dialysis. In primary peritonitis, previous medical history and medications are quite suggestive. Secondary peritonitis may follow any abdominal infection such as appendicitis, perforation of hollow viscus, typhoid or tubercular ulcer of gastrointestinal tract, pancreatitis, inflammatory bowel disease, pelvic inflammatory disease, urinary tract infection or abdominal trauma etc. The paramount concern in management is to relieve the patient from the toxins and bacterial loads from the abdominal cavity and control of the source as early as possible. Any improper evaluation and interventional delay might have a fatal outcome. The patient should be hospitalized immediately and treated with broad spectrum intravenous antibiotics, parenteral fluid, and nutritional support. Commonly encountered surgical indications of peritonitis include ruptured appendix, perforated duodenal ulcer, colonic diverticulitis, typhoid ulcer perforation, ischemic colitis, pelvic inflammatory disease and so on .Emergency exploration is indicated to control the source of sepsis even in diagnostic uncertainty. In this regard, the value of laparoscopy has been recognized since 1950. Laparoscopy under local anaesthesia is also useful in acute peritonitis to visualize the whole abdomen and pelvis providing the diagnosis which might be missed in conventional imaging ,5. Even This is a report on a single patient with etiological uncertainty and its outcome of interest with laparoscopic surgery. We managed our patient in Jahurul Islam Medical College Hospital, a tertiary care center at a remote area in February 2019. This work was reported in line with the SCARE criteria [2A 32-years-old man was admitted with the complaints of severe and diffuse abdominal pain associated with mild fever for last 8 h. He had no bowel movement within last 24 h and respiratory discomfort for one and half an hour. He was hemodynamically stable with optimum oxygen saturation and temperature was recorded 99 °F with some signs of dehydration. His abdomen was not distended but it was tender all over and rigid and digital rectal examination revealed normal. Treatment started with intravenous crystalloid infusion with antibiotics (cefoperazone + metronidazole) and analgesics.Plain chest and abdominal radiograph with abdominal ultrasound  appearedhttps://youtu.be/DvZvXl20-ns). Anesthetic recovery was uneventful. The patient had symptomatic improvement on first post-operative day (POD) and DT collection was noted 150 mL, which gradually declined. Bowel sound appeared on 3rd POD and oral feeding resumed. Peritoneal fluid culture revealed no growth. Histopathology of the appendix revealed acute appendicitis. He was discharged with DT on 4th POD and did not develop any further complications like recurrent abdominal pain, intestinal obstruction, or any abdominal sepsis within 1 year of post-operative follow-up.Diagnostic laparoscopy proceeded with modified direct initial trocar access through umbilicus as our routine practice and other ports were placed as needed . Huge am3Exploratory Laparotomy has been the mainstay of surgical management in acute peritonitis. And laparoscopy had been considered relatively contraindicated because of poor vision, inadequate peritoneal toileting, increased sepsis, time-consuming, post-operative adhesions, and late complications. But the recent trend is inclining more towards the laparoscopic management in acute peritonitis. We shall discuss the diagnostic dilemma and laparoscopic management outcome with our case report.The medical history of our patient was not suggestive of primary peritonitis. The accurate diagnosis was crucial because many abdominal pathologies may mimic a similar presentation. Lactate, C-reactive protein are the well-known biomarkers of inflammation and raised in acute abdomen. But these are nonspecific and may mislead the diagnosis and require other unnecessary invasive interventions . The con“…. there are no contraindications to create pneumoperitoneum when laparoscopic surgery is applicable in cases of peritonitis (grade B).” The results of animal studies regarding the influence of pneumoperitoneum on bacteremia and endotoxemia are controversial [Argesta F. et al documented 95% success rate with laparoscopic intervention underwent within 48 h of admission over 935 patients with acute peritonitis [A few inconsistent data mentioned increased abdominal pressure (IAP) during pneumoperitoneum seems to promote bacteremia . But difoversial . Timely itonitis . In our The practical difficulties we used to encounter with laparoscopy in generalized peritonitis are difficult torcher access due to distended bowel loops, previous surgical scars, and peritoneal adhesions, and the possibility of iatrogenic injury, etc. Early conversion to open is essential in difficult cases. Laparoscopic management in patients with acute peritonitis recorded up to 23.3% conversion rate in a research article and was mainly due to intra-abdominal adhesions, obscured anatomy, and iatrogenic lesions .Acute appendicitis is a disease mostly diagnosed clinically, yet investigations are advised accordingly to exclude other differential diagnoses. And any specimen retrieved from the abdomen with generalized peritonitis is expected to be infiltrated with acute inflammatory cells. Histologic appearance of acute appendicitis is obvious in this case but not seemed to be the etiology unless the appendix was gangrenous or perforated ; E, G. S4Clinical suspicion, the actual pathology, and the ultimate intervention are always subject to vary in acute abdominal conditions. In a suspected case of secondary peritonitis, laparoscopy could be attempted with the preparation of conversion to have the most accurate diagnosis and the best therapeutic intervention. And simultaneously the hazards of an unnecessary laparotomy could be avoided.No conflict of interest.Solely funded by the authors.This case reports the outcome of a routine management. Ethical Committee approval was not taken.Informed written consent was taken for scientific publication and presentation.1. Name of the registry: n/a2. Unique identifying number or registration ID: n/a3. Hyperlink to your specific registration (must be publicly accessible and will be checked): n/aDr. Md. Sumon Rahman.Not commissioned, externally peer-reviewed.Md. Sumon Rahman: Conceptualization, Methodology, Data curation, Writing - original draft, Funding acquisition, Supervision, Validation. Hasan Ul Banna: Formal analysis, Writing - review & editing. Md. Nazmul Hasan: Data curation, Software. Mohammad Jumman: Data curation.


Giant hydronephrosis is defined as a dilated collecting system containing more than one liter of fluid. The diagnosis of giant hydronephrosis is rare due to improved diagnostics and the liberal use of abdominal imaging. Herein we report a 40-year-old woman who presented with acute onset abdominal pain and was diagnosed with giant hydronephrosis. She underwent a simple open nephrectomy and made an unremarkable recovery. Although giant hydronephrosis due to ureteropelvic junction obstruction is common in the pediatric and adolescent age group, it rarely presents in adults. Acute presentations, like abdominal pain, are exceedingly rare. Judicial use of cross-sectional imaging, as in our patient, can confirm the diagnosis and help in successful management. Idiopathic obstruction at the ureteropelvic junction (UPJ) is a major cause of obstructive uropathy at all ages and is often detected and treated in early childhood. It is a spectrum of pathophysiological processes of congenital  and acquired  etiologies . The incA 40-year-old woman presented to the casualty of our tertiary care hospital with acute onset left loin pain and dyspnea of one-day duration. The pain was not radiating and was felt predominantly over the left loin and lumbar regions. There was no previous history of hematuria or urolithiasis. She denied any history of trauma or previous abdominal surgeries and did not suffer from any comorbid illness. She was afebrile, and the rest of her general examination was unremarkable. Vital signs like pulse rate and blood pressure were normal, while the abdominal examination revealed a localized bulge in the epigastric, umbilical, left lumbar, hypochondriac, iliac fossa, and renal angle regions on inspection. Palpation confirmed a tender, firm, vague mass that moved with respiration. The mass was bimanually palpable and ballotable clinically, indicating a renal mass. The renal angle was tender on palpation, but there was no other evidence of sepsis. Findings of an examination of the respiratory and cardiovascular system were normal. Her laboratory evaluation results, such as renal function tests, electrolytes, complete hemogram, urine microscopy, and urine culture, were within reference limits. Findings from her chest roentgenogram, electrocardiography, and echocardiography were also normal. An ultrasonogram of the abdomen revealed a 26 cm x 20 cm x 15 cm hydronephrotic left kidney pushing the bowel and other solid organs to the right side  of the abdomen was done, which revealed a grossly enlarged left kidney (measuring 30 cm x 20 cm x 18 cm) with no uptake, excretion, or drainage of contrast into the collecting system (Figure In light of the giant hydronephrotic nonfunctioning left kidney, a left-sided open simple nephrectomy was performed through a left eleventh rib flank incision using the extrapleural and extraperitoneal approach after informed consent. The giant hydronephrosis crossed the midline pushing the bowel to the opposite side and reached inferiorly as far as into the pelvis Figure . It was Giant hydronephrosis is the presence of more than one liter of fluid in the collecting system of a hydronephrotic kidney. Although it is classically caused by UPJO, other conditions such as ovarian cysts, pancreatic cysts, hydatid cysts, and large adrenal cysts can mimic this peculiar condition of the kidney . With thDiagnosis of giant hydronephrosis is usually made preliminarily by ultrasonogram, which demonstrates the giant fluid-filled hydronephrotic sac and thinned out renal parenchyma. However, cross-sectional images and CECT with delayed images are needed to ascertain the functional status of the hydronephrotic kidney. Contrast uptake, excretion, and drainage in the collecting system can be assessed . MRI is The treatment of giant hydronephrosis depends on the functional status of the kidney and underlying etiology. Nephrectomy is the procedure of choice for nonfunctioning kidneys, and pyeloplasty is the surgical management of UPJO with good differential function . PercutaThis case has been presented for its rarity and to emphasize the fact that although UPJO is a rare diagnosis in adults presenting with an acute abdomen, it can still be detected as late as the fourth decade of life in developing and underdeveloped countries. Therefore, a high clinical suspicion is required for clinicians to diagnose this unexpected entity among adults with acute onset abdominal pain.


Small bowel stenosis after blunt abdominal trauma is relatively rare, and progression from trauma to bowel stenosis might sometimes be delayed. Herein, we report the case of a patient who was diagnosed with small bowel stenosis relatively early and received laparoscopic surgery.An 18-year-old Japanese male was in a traffic accident and was urgently transported to our hospital. On arrival, he was admitted with right kidney and right adrenal injury and abdominal aortic aneurysm. On hospital day 13, he vomited during conservative treatment without surgery, and computed tomography revealed small bowel stenosis and dilatation of the oral-side small bowel. No improvement with the ileus tube occurred, and he received laparoscopic surgery on hospital day 21. Briefly, the abdominal cavity was observed with a laparoscope. The mesentery was congested, scarring around the stenotic small bowel regions was present, and three stenotic regions were observed 40–50 cm from the Treitz ligament. The patient received partial resection and anastomosis of the small bowel. The postoperative course was stable, and he was discharged on postoperative day eight.Most cases of bowel stenosis after abdominal trauma are irreversible and usually require surgical treatment. Therefore, small bowel stenosis should be considered in patients with abdominal symptoms after blunt abdominal trauma. Gastrointestinal damage due to blunt abdominal trauma often presents with symptoms immediately after injury due to organ rupture or bowel perforation and requires emergent surgery. However, patients rarely develop small bowel stenosis or obstruction several days to months after the initial trauma. Herein, we report the case of a patient with small bowel stenosis on day 13 after blunt abdominal trauma who was successfully treated with partial small bowel resection by laparoscopic assistance.An 18-year-old Japanese male patient was urgently transported to our hospital following a traffic accident. The patient was sitting in the passenger seat with the seat belt fastened and had a car-to-car accident. The patient was diagnosed with right kidney injury, right adrenal injury, and abdominal aortic aneurysm and was admitted to the hospital. Intra-abdominal hemorrhage due to renal injury was controlled by endovascular treatment. The abdominal aortic aneurysm was followed without any intervention. Other organs, including the small bowel, did not show any damage on the first computed tomography (CT) examination. The clinical course of the patient was good after these treatments, and food intake was initiated on the tenth hospital day. However, on hospital day 13, the patient vomited bile-like fluid twice. He experienced no abdominal pain, and hematological tests did not reveal any abnormal findings. CT showed stenosis and oral-side dilatation of the small bowel Fig. . Based oFirst, a 12-mm port was placed using the open method in the umbilical site and the abdominal cavity was observed with a laparoscope. A localized area of the mesentery was congested and edematous, and scarring around the stenotic section of the small bowel was present Fig. a, b. A sThe excised specimen showed three stenotic regions Fig. . The patThe postoperative course was stable, and the patient was discharged on postoperative day eight.Patients with gastrointestinal damage due to blunt abdominal trauma often present with acute symptoms due to bowel rupture and/or bleeding, usually requiring emergent surgery. However, in rare cases small bowel stenosis or obstruction occurs several days or months after the trauma –3. The uIn our case, we performed laparoscopic surgery. If the surgery needs to be performed within a short period after the injury, open surgery may be chosen because of the possibility of damage to other organs and the need for a detailed intra-abdominal search. However, in our case, it had been several days since the injury, and a diagnosis of only small bowel stenosis was made, with no other abnormalities; furthermore, decompression of the small intestine was successful using an ileus tube. Therefore, we selected laparoscopic surgery. The 15-year-old patient underwent partial resection of the small intestine with a 3-cm incision; it was less invasive, and laparoscopic surgery was also useful in terms of integrity.Bowel stenosis due to abdominal trauma is often located near the body midline such as the upper small bowel and terminal ileum, where the mesentery is relatively fixed –8. ExcepWe searched the literature from 1980 to 2018 using the keywords “abdominal trauma,” “late onset,” and “small bowel stenosis” using the PubMed and the Japan Medical Abstracts Society electronic databases, and the 62 collected cases were reviewed, 47 males and 15 females with an average age of 40.5 (5–80) years. The cause was traffic accidents in 41 of these 62 cases. In 87% of the cases, the location of delayed small bowel stenosis was within 100 cm from the ileocecal region and within 200 cm from the Treitz ligament (Fig. Several reasons underlie delayed diagnosis and surgery in these patients. First, temporal conservative treatment might improve symptoms in patients with incomplete stenosis. Additionally, since symptom development takes time, the patient might be handled without the knowledge of prior blunt abdominal trauma if they are treated for stenosis-related symptoms in a hospital other than the emergency hospital handling the initial trauma. Therefore, obtaining detailed medical history on abdominal trauma is important to ensure early diagnosis.Post-traumatic intestinal stenosis should be considered in patients with a history of blunt abdominal trauma presenting with abdominal symptoms.


Protein-losing enteropathy and spontaneous isolated superior mesenteric artery dissection are both rare clinically. Protein-losing enteropathy due to superior mesenteric artery dissection is extremely rare.A 46-year-old male with acute abdominal pain and hematochezia was diagnosed with a complete occlusion of the superior mesenteric artery because of dissection. He suffered from diarrhea and hypoproteinemia after an emergency thromboendarterectomy.Based on laboratory tests and capsule endoscopy inspection, a diagnosis of protein-losing enteropathy was made.Endovascular treatment was provided.After stent placement, he quickly recovered without a recurrence of symptoms.Protein-losing enteropathy is a serious complication of an isolated superior mesenteric artery dissection. Restoring the patency of the superior mesenteric artery is keyed for the treatment of this complication. A disorder associated with the superior mesenteric vessel is a rare cause of PLE.,4Protein-losing enteropathy (PLE) is a rare clinical condition characterized by the excessive loss of plasma protein into the gastrointestinal tract resulting in hypoproteinemia. Here, we present a case who suffered from PLE, resulting from the postoperative SISMAD, and who recovered after endovascular treatment. The patient provided informed consent for this publication.Spontaneous isolated superior mesenteric artery dissection (SISMAD) is also regarded as a rare condition. To the best of our knowledge, PLE as a complication of superior mesenteric artery dissection (SMAD) has been rarely reported.29/L (4.0–10.0 × 109/L) and normal albumin level: 42 g/L (35–50 g/L). Two hours after admission, he underwent an emergency exploratory laparotomy because of persistent abdominal pain and hematochezia. During the operation, the entire small intestine was pale and no palpable pulses in the SMA trunk could be detected. The true lumen was completely compressed by the thrombotic false lumen. A proximal thrombectomy and endarterectomy were performed, and the distal intimal flap was fixed to the wall of the SMA. The pulsation of the SMA and the color of the small intestine were restored after this process.A 46-year-old man, who presented with a 4-h history of acute abdominal pain, nausea, vomiting, and hematochezia, was admitted to our emergency department. He had no history of trauma, abdominal pain, etc and his blood pressure is normal (125/76 mm Hg). Contrast-enhanced computed tomography angiography (CTA) showed that the superior mesenteric artery (SMA) was completely occluded due to dissection . Although the diarrhea symptoms were controlled to 3 to 5 times a day by conservative treatment, his serum albumin level began to continuously drop. He was given a vasodilator, albumin, and nutritional support through intravenous infusion every day to avoid severe hypoproteinemia. After 30 days, his albumin level fell to around 13 to 18 g/L. The CTA showed that the branches of the SMA were patent but the SMA trunk was occluded by about 1 cm Fig. . Capsule3–4PLE is a rare cause of hypoproteinemia in the clinic. In healthy individuals, protein loss through the intestinal epithelium plays a tiny role in total protein loss. If no other causes of hypoproteinemia can be identified, then diagnosis of PLE should be considered. The clinical manifestations of PLE are variable, which are related to the underlying cause. However, hypoproteinemia contributes to the primary symptoms, such as edema and malnutrition. Choosing the best treatment for SMAD is not definitive thus far. Most patients can be managed by conservative treatment. However, emergency surgery or endovascular treatment is necessary for the patients suffering from impending arterial rupture or bowel gangrene.Only a few cases of SMAD have been reported previously, but the cases in China have significantly increased in recent years.For this patient, the occlusion of SMA after the operation was perhaps because the swollen flap was lifted by blood flow to form a valve that obstructed the blood flow. Fortunately, the collateral branch avoided the necrosis of the intestine. However, the blood supply of the collateral circulation was apparently not sufficient and the bowel was in a chronic ischemia condition, which led to the erosion of the mucosal epithelium. The intracellular permeability increased and tight junctions between cells became wider. The erosions of the intestine mucous membranes had been shown by the capsule endoscopy. Inevitably, the continuously protein-rich fluid loss from the eroded epithelium to the gastrointestinal tract would lead to diarrhea and hypoproteinemia.PLE caused by SISMAD is rare. Refractory hypoproteinemia is a clinical feature of this disease. This case had a history of SMAD dissection, and CT showed segmental SMAD occlusion, indicating the possibility of ischemic lesions in the intestine. Capsule enteroscopy confirmed the segmental ischemic erosion of the small intestinal mucosa. The above features support the diagnosis of PLE. In addition, a small intestinal mucosa biopsy or (99 m) Tc-labeled albumin imaging can help in the diagnosis, but the latter is not often used clinically. Regarding the reported cases of PLE caused by mucosal erosion, many of them underwent enterectomy of the pathological segment.–4 The cause in this case was intestine ischemic due to SMAD, and the first vascular reconstruction had not completely corrected the intestinal ischemia. Therefore, endovascular therapy is preferred to restore blood flow.,6 The balloon-expandable stent was chosen because it was easy to locate and does not cover more branches. After the stent placement, the erosion of the mucous could be repaired slowly and the serum albumin increased continuously. Therefore, for these types of postoperative patients with chronic intestinal ischemia, endovascular treatment is a simple, safe, and effective optional measure.Treatment options for PLE should be directed at the underlying condition. However, if edema, ascites, malnutrition, or micronutrient deficiency is present, intravenous albumen supplement and nutritional treatment are necessary.Conceptualization: Xicheng Zhang.Data curation: Yuan Sun.Investigation: Yuan Sun.Supervision: Dengqiu Zhao.Validation: Zhaolei Chen.Writing – original draft: Xicheng Zhang, Dengqiu Zhao.Writing – review & editing: Xicheng Zhang, Yuan Sun.