Benign tumours of vascular and lymphatic origin are known as lymphangiomas. In this report, we present a case of a 26-year-old lady admitted with symptoms of small bowel obstruction. Her computed tomography (CT) scan showed a well-defined mass in the small bowel mesentery associated with small bowel volvulus. Segmental resection of the bowel, including the mass, was performed. Microscopic examination and immunohistochemistry of the specimen were consistent with lymphangioma of the small bowel mesentery. Lymphangiomas are benign tumours arising from the vascular and lymphatic origin . They arSmall bowel volvulus is one of the causes of bowel obstruction as a result of torsion of the small bowel and its mesentery. It is a well-recognised condition that might affect infants and children; however, it appears to be a rare disease in adults . In thisA 26-year-old lady presented to the accident and emergency (A&E) department with a 24-hour history of left-sided abdominal pain. There was no associated nausea or vomiting. She also reported opening her bowels 24 hours prior to the presentation. She was a smoker (seven to 10 cigarettes per day) but denied alcohol consumption. Upon questioning, she also gave a three-month history of vaginal bleeding following a medically induced abortion.On clinical examination, she had abdominal distension and tenderness confined to the left iliac fossa and left flank. Her blood tests showed leucocytosis; however, her liver function tests (LFTs), urea and electrolytes (U&Es), amylase, C-reactive protein (CRP), and lactate were unremarkable.A computed tomography (CT) scan of the chest, abdomen, and pelvis was arranged and showed a lobulated well-defined low-density mass in the mesentery measuring approximately 7 cm in diameter and was associated with a number of prominent vessels that extended into the matrix of the lesion. There was also an associated small bowel volvulus with compression of the superior mesenteric vein (SMV) at the level of the mesenteric root Figures -4.After discussion and appropriate consent, the lady was taken to theatre for diagnostic laparoscopy which revealed a small amount of chylous fluid in the pelvis associated with a proximal jejunal mesenteric mass and scattered mesenteric rubbery lymph-nodes. The mass was causing a proximal mesenteric twist. Moreover, it was tethered to the root of the mesentery so laparoscopic mobilisation was unachievable. Therefore, conversion to open surgery was performed uneventfully using a midline laparotomy incision. The twist was undone and the small bowel jejunal segment was excised together with the whole mass and side to side primary anastomosis was performed using a linear stapler.The postoperative period was uneventful. She improved steadily over a few days. She was able to tolerate oral intake and opened her bowels. Consequently, she was discharged on the fifth postoperative day with a planned follow-up in eight weeks’ time.Microscopic examination of the specimen showed a vasoformative lesion within the small bowel mesentery, composed of dilated, thin-walled channels and cystic spaces lined by cytologically bland, flat endothelial cells and containing proteinaceous fluid along with small lymphocytes and a few red blood cells. Some of the vessel walls contained bundles of smooth muscle. Small lymphocytes were present throughout the septa separating the vessels, with the formation of numerous lymphoid follicles. The lesion expanded the mesentery, extending from just beneath the serosa up to the muscularis propria focally. The separately sampled lymph nodes showed reactive changes. On immunohistochemistry, the endothelial cells were positive for podoplanin (D2-40) and factor VII. Immunostaining for human melanoma black-45 (HMB45) and calretinin was negative. Further stains showed diffuse positivity with a cluster of differentiation-13 (CD31) within the endothelial cells, actin, desmin, calponin, and caldesmon highlight bundles of smooth muscle in the vessel walls. AE1/AE3 cytokeratins and cytokeratin 5/6 (CK5/6) were negative. A diagnosis of lymphangioma of the small bowel mesentery was concluded and a referral to the sarcoma multidisciplinary team (MDT) meeting was made. After the MDT discussion, it was agreed that there was no evidence of sarcomatous changes.Unfortunately, because of repeated appointment cancellations by the patient, her follow-up was not possible and she was discharged back to her general practitioner (GP).Cysts of the mesentery are rare. It was first described by Benevieni in 1507, yet it was not until 1884 when Rokitanski described a chylous cyst of the mesentery . They arApproximately 95% of lymphangiomas are found in the neck and axilla and the other 5% occur in the mediastinum and abdominal cavity including the mesentery, retroperitoneum, and bones . When loThe aetiology of lymphangiomas is unknown. Multiple theories have been found including embryonal origin, retention, endothelial secretory disruption, inflammatory, lymph node degeneration, trauma, surgery, and radiation therapy ,5. WegneHistologically, lymphangiomas are differentiated from mesenteric cysts by the presence of endothelial lining, connective tissue, and smooth muscle fibres in the former and the cuboidal or columnar lining with a lack of smooth muscles in the latter . ImmunohMesenteric lymphangioma may be discovered incidentally or manifests as intestinal obstruction, volvulus, intussusception, ischemia, mass effect, gastrointestinal bleeding resulting in anaemia, or rupture and shock ,6,7,9,10The definitive diagnosis of mesenteric lymphangioma is made by histopathological examination, nevertheless, imaging may aid in the diagnosis. Sonographic examination classically shows an anechoic cyst with multiloculated and occasional echogenic debris . ComputeSmall bowel volvulus is a rare condition among adults and is usually classified into two categories . The priThere are two theories explaining the relation between small bowel volvulus and lymphangioma. It was argued that the flaccid and mobile nature of a mesenteric lymphangioma can result in its rotation which induces small bowel volvulus. The other theory implies that longstanding or intermittent volvulus causes lymphatic obstruction which results in lymphatic cysts. These cysts are generally unilocular cystic masses without internal septations .While there have been reported cases of spontaneous regression in size of mesenteric lymphangioma, management is mainly surgical aiming at complete surgical resection whenever feasible even in asymptomatic patients, however, some advocate follow up and surveillance in incidentally discovered cases ,6,11. SuIn this study, we presented a case of mesenteric lymphangioma associated with small bowel volvulus in an adult. Complete surgical excision along with the affected small bowel segment was achieved through laparotomy. After the histopathological and immunohistochemical assessment, the diagnosis of mesenteric lymphangioma was established. We recommend follow up with imaging such as abdominal ultrasound, CT, or MRI scans as recurrence might occur.


Gastrointestinal stromal tumors represent the most frequently encountered primary mesenchymal tumors. Whereas the liver and the peritoneum are known to be the preferential metastasis sites, no therapeutic standard has yet been established for the management of bone metastases because of their very low incidence. We report a unique example of a single humerus metastasis of a jejunal gastrointestinal stromal tumor.We report the case of a 72-year-old European woman whose jejunal gastrointestinal stromal tumor was resected in 2013 and treated during the following 3 years with imatinib (400 mg daily). In 2018, she developed a single humeral bone lesion that was identified as a gastrointestinal stromal tumor metastasis. After 7 months of imatinib intake, reconstructive surgery was performed. Pathologists confirmed the satisfactory histological regression and assessed the complete tumor resection. The patient is still on imatinib maintenance therapy, with no recurrence reported so far. She fully recovered the upper limb function after following an appropriate rehabilitation program.Current literature and published case reports indicate that bones are one of the rarest locations of gastrointestinal stromal tumor metastasis (about 1%), with occurrence mainly in the spine. Patients initially diagnosed with gastrointestinal stromal tumor of the small intestine and stomach are more likely to suffer from bone metastasis, compared with other gastrointestinal stromal tumor locations. The median overall survival rate is higher for patients with isolated bone metastasis compared with those having liver metastasis. Metastasis occurs on average 4 years after the primary, but it may take up to 20 years, emphasizing the need for long-term clinical and radiological monitoring. Although specific guidelines for such cases have not yet been established, we suggest that a multimodal concerted approach involving surgery or radiotherapy associated with tyrosine kinase inhibitor intake should be considered.Bones are one of the rarest locations of gastrointestinal stromal tumor metastasis. A multidisciplinary collaboration was set up to allow conservative surgery of our patient after several months of imatinib treatment. A year and a half later, the patient is still in complete remission. This specific case supports the concept of an intermediate stage between local and oligometastatic disease that should be managed with a curative aim, as much as possible. Gastrointestinal stromal tumors (GIST) are the most common mesenchymal tumors of the gastrointestinal tract. The incidence rate varies from 15 to 30 cases per million people, and keeps increasing [Interstitial cells of Cajal in myenteric plexus are known to be the precursors to these mesenchymal tumors. GIST can involve any location in the gastrointestinal tract but are most frequently encountered in the stomach and small intestine , 4, 5. AComplete resection remains the best option for localized tumors. Health care guidelines and prognosis rapidly changed after tyrosine kinase inhibitors (TKI) were brought to the market.With modern targeted therapy, patient overall survival has increased, and the course of GIST diseases keeps evolving, as documented in the literature, requiring constant adaptation of patient care and therapeutic follow-up . PrevaleWe report the medical history of a 72-year-old woman who developed a single humeral bone metastasis 5 years after complete surgery of the primary tumor.The 72-year-old Mrs T. of European origin consulted her physician because of symptoms of abdominal distension, dyspepsia, and anorexia, which caused a 5 kg weight loss. Her personal medical history consisted of drug-induced chronic pancreatitis and a resected benign breast nodule. She stated that she had no allergies and no smoking habits. Chronic medication consisted of a daily pancreatic enzymatic substitution. The patient was professionally active as an employee in an insurance company, with no relevant exposure reported. Her family history included a case of colorectal cancer and of breast cancer, for an aunt and a cousin, respectively.Radiological examinations reported a mobile pararectal mass at least 8 cm long, without any other lesion. Blood markers  levels were in the usual range. Initial explorative celioscopy in August 2013 was subsequently converted to laparotomy, which allowed complete resection of a jejunal tumor. No other abdominal nor pelvic lesion was detected. Pathological analysis confirmed the diagnostic of a GIST with typical immunohistochemical profile and strongly positive DOG1, CD34, and CD117 markers. The Ki67 proliferation index was evaluated at 10%, and the necrosis contingent was below 50%. Molecular sequencing revealed that the exon 11 of C-KIT gene was deleted (as in 70% of GIST) [2, it was classified as belonging to the 6b group in the Miettinen classification [Considering that the tumor was located in the intestine, that its largest dimension was 11 cm and its mitotic index higher than 5 per mmfication . ConsequNo clinical event happened until October 2018, when the patient reported shoulder and left-arm painful functional limitation. Humeral bone swelling was clinically palpable. Brachial magnetic resonance imaging (MRI) and computed tomography (CT)  classification and the Miettinen classification are based on tumor size, mitotic count, and location . On the Whereas bones and lung have also been found to be susceptible to host metastasis –13, no mOn the basis of literature data, the percentage of patients developing bone metastasis varies from 0.47 to 5.5% , with spMetastasis occur on average 4 years after diagnosis of the primary GIST, but can take up to 20 years to occur . Thus, oWe proposed a complete resection of the humeral metastasis, framed by a targeted therapy with imatinib, although standards of care recommendations are not yet established, because such cases have only rarely been reported . After hMoreover, the development of well-known hepatic metastasis during evolution of colorectal cancer requires, for the patients considered eligible, an aggressive oncological and surgical approach to improve overall survival , 19. TheRecently, other TKI such as avapritinib or ripretinib appear to be interesting for fourth-line therapy or in cases of specific secondary resistance mutations , 21. It No specific molecular profile among those identified  appears Our knowledge of the evolution of diseases such as GIST is progressing, as witnessed in the current literature, and thus the need for medical oncologists to implement long-term patient surveillance, with modalities depending on the initial recurrence risk . This reIn conclusion, appropriate clinical and surgical management of such clinical cases requires active multidisciplinary collaboration between medical oncologists, radiotherapists, surgeons, and pathologists to achieve optimal overall survival and patient quality of life.


Teaching point: When the joints between bones are ankylosed, such as in ankylosing spondylitis, Paget’s disease can involve multiple contiguous bones leading to pagetic vertebral ankylosis. A 76-year-old male was referred to our radiology department to perform an abdominal computed tomography (CT) as part of the follow-up after Whipple surgery for pancreatic intraductal papillary mucinous neoplasm. He had a prior history of Paget’s disease (PD) and ankylosing spondylitis (AS) and was totally asymptomatic.Figure 1), iliac bones , and costovertebral joints (not shown). MRI demonstrated a normal fatty bone marrow signal consistent with uncomplicated PD in the late or inactive state (not shown).CT showed characteristic features of AS  associated with typical bone remodeling features of PD  resulting in an extensive pagetic vertebral ankylosis (PVA) with involvement of the dorsolumbar spine, sacrum (PD is a chronic condition characterized by constant bone remodeling involving osteoclastic and osteoblastic mechanisms. The most common sites of involvement are pelvis, lumbosacral spine, femur, or tibia and at least 50% of patients present with polyostotic disease .Usually, PD does not progress to adjacent bones as the soft tissues act as natural barriers.Some authors suggested a possible progression of the pagetic process through discs that presented signs of degeneration, but it remains unclear if intradiscal pagetic invasion is possible through healthy disc tissue. Otherwise, there is evidence of PVA in patients suffering from PD and a disease responsible of bone bridging such as AS or diffuse idiopathic skeletal hyperostosis. This case report suggests another hypothesis for the progression of the pagetic process to contiguous bones through syndesmophytes or osteophytes, leading to PVA .In our case, there is a continuum of PD bone remodeling involving iliac bones, sacrum, thoraco-lumbar spine, and ribs. The extension through adjacent vertebral bodies is explained by the presence of syndesmophytes and the extension through iliac bones and ribs might be explained by the fusion of the sacro-iliac and costo-vertebral joints secondary to the AS inflammatory process.Our findings support the hypothesis that PD may gradually progress in the contiguous ankylosed bones through acquired bone bridges and explain one of the most extensive PVA cases reported so far.


MAP2K1 along with a brief review of the literature.Syringocystadenocarcinoma papilliferum (SCACP) is a rare malignant neoplasm arising from adnexal tissues and is the malignant complement to the benign neoplasm syringocystadenoma papilliferum (SCAP). SCACP lesions appear as raised nodules or inflammatory plaques and can be associated with SCAP or nevus sebaceous. There have been fewer than 100 described cases of this neoplasm in the literature, and all previously published cases have been described in adults, with the majority occurring in the elderly. We present a case of an adolescent female with a syringocystadenocarcinoma papilliferum arising from a large thigh mass harboring an in-frame alteration in  A fifteen-year-old female presented to our institution with a large pedunculated posterior thigh mass. According to the patient's mother, the lesion initially appeared as “three small cuts” at birth and grew proportionally with the patient, now appearing hypervascular and measuring 11.5 × 6 × 2 cm in size . There wThere was no prior imaging. An ultrasonographic examination demonstrated two hypoechoic masses with Doppler flow showing arterial and venous enhancement. A subsequent MRI showed the extent of the masses to be 11.9 cm × 6.3 cm × 1.8 cm, extending down to muscle fascia with robust perfusion supplied by perforators of the deep femoral artery . The patMicroscopic examination revealed an exophytic, papillary lesion with an abrupt transition from the skin surface, overlying a tubular and cystic component diffusely involving the dermis . Papilla2 were present throughout the lesion. CK7 immunohistochemistry was diffusely positive in the epithelial cells of the exophytic component and luminal epithelial cells in the dermal component  (chest) and magnetic resonance imaging (MRI) (abdomen/pelvis), as well as a full-body PET scan, were obtained. No distant metastases were identified. To complete staging, a left inguinal sentinel lymph node biopsy was performed in conjunction with wide reresection using 2 cm margins. Reresection included all subcutaneous tissue to the level of the semimembranosus muscle fascia in order to ensure complete resection. A skin graft was required to close the large defect on the posterior thigh, which healed well without complication . AlthougMAP2K1  resulting in an in-frame deletion of codons Leu101-Lys104 (ΔLEIK), replaced with two amino acids (Asn and Ser) (c.301_311delinsAATTC:p.Leu101_Lys104delinsAsnSer) (β3-αC loop within the kinase domain (encoded by amino acids 98–104) encompass a class of MAP2K1 variants that demonstrate RAF-independent constitutive signaling due to receptor homodimerization and transphosphorylation.Given the rare diagnosis of SCACP, the patient was enrolled on an institutional translational research protocol . PairedsAsnSer) . This insAsnSer) . AlthougsAsnSer) . In-framThis patient is the youngest described case of SCACP in the literature, which reports the median age of SCACP to be 63.6 years (reported range 22–93) . These lWide local excision is the treatment of choice for SCACP, though there have been cases where SCACP was treated effectively with chemotherapy and radiation in patients with inoperable tumors or who are poor surgical candidates . SpecifiPathologic examination of SCACP resembles SCAP. In both variants, the epithelium is typically composed of an inner columnar cell layer and an outer cuboidal layer. The stroma of the tumor often contains plasma cells and other inflammatory cells . In SCACHistologically, SCACP can vary widely in presentation, ranging from an adenocarcinoma-like appearance to having features more consistent with invasive squamous cell carcinoma . This hiHRAS p.Gly13Arg and KRAS p.Gly12Asp was first described in nevus sebaceous, prompting additional study in secondary tumors arising from this lesion. BRAF p.Val600Glu was later identified in a subset of SCAP. In this case report, we expand the genomic landscape to include activating alterations within the MAP2K1 gene, as the MAP2K1 p.Leu101_Lys104delinsAsnSer is predicted to activate RAS-MAPK signaling. Given the clinical utility of MEK inhibitors in tumors with described RAS-MAPK pathway activation and ongoing clinical trials, the utility for targeted inhibition in patients with in-frame MEK1 β3-αC loop alterations has been explored. Notably, allosteric MEK inhibitors (MEKi), such as trametinib, are ATP noncompetitive kinase inhibitors that bind and stabilize an inactive conformation of MEK1. MAP2K1 missense and in-frame deletions occurring outside of the β3-αC loop (amino acids 98–104) demonstrate full or partial dependence on RAF activation and exhibit sensitivity to allosteric MEKi. However, in-frame deletions within the β3-αC loop, such as described in this report, result in RAF-independent stability of the inactive MEK conformation and resistance to allosteric MEKi. A selective ATP-competitive MEKi, MAP855, has shown promising results through inhibition of pERK across all MAP2K1 alterations and may be of future benefit to patients with tumor harboring in-frame β3-αC loop deletions.The genetic landscape of SCACP is poorly characterized; however, aberrant signaling of the RAS-MAPK pathway has been associated with benign precursors, including nevus sebaceous and SCAP. Mosaicism for In conclusion, SCACP is a rare malignancy often arising within an existing nevus sebaceous or SCAP in adults. Our case report demonstrates that SCACP can also rarely affect pediatric patients. Awareness of such atypical presentations can facilitate timely diagnosis. Standard clinical management guidelines for surgical management and surveillance are lacking, and future studies outlining strategies for clinical management are desired.


Female Genital Mutilation/Cutting (FGM/C) is the practice of cutting parts of the female external genitalia in fulfillment of sociocultural obligations and in some cases for nonmedical reasons. It is classified into 4 main types depending on the extent of cutting. Some forms of FGM/C are common in at least 29 countries globally, mainly in Africa. The overall prevalence of FGM/C in Ghana is approximately 4%. The motivation for this practice varies from community to community but includes the fulfillment of cultural values, uplifting the girl child, and, according to some reports, reducing sexual desire and promiscuity. The objective of this article is to illustrate how FGM/C resulted in sexual dysfunction in a young woman married for 2 years. We present a 19-year-old female who was subjected to female genital cutting in her formative years who presented with apareunia for 2 years in her marriage. We illustrated how FGM/C led to a genital tract obstruction with resultant sexual dysfunction. Examination revealed a Type 3 FGM/C (infibulation) with almost complete occlusion of the genital tract. She underwent a successful defibulation and resumed sexual activity with her husband within 6 weeks of the procedure. Female Genital Mutilation (FGM) is the term used by the World Health Organization (WHO) to refer to all medically unnecessary cutting of the external female genitalia, often carried out to fulfill perceived sociocultural obligations , 2. ThisYoder et al. reported that over 100 million women have been affected by FGM/C globally and a further 3.3 million girls are affected yearly . Some foA 19-year-old circumcised girl from Northern Ghana presented to the hospital with complaints of apareunia since she got married 2 years before presentation. She had no lower abdominal pain, vaginal discharge, or fever. She had no past medical or surgical history. She had her menarche at 14 years and had regular monthly cycles, with 7-day flow, and no dysmenorrhea or menorrhagia. She had not had any formal education. She and her partner are farmers.On examination, she was stable, not pale, and looked well nourished. Normal blood pressure readings were obtained. Her breast was well developed. The abdomen was flat, soft, and nontender with no masses palpated. Examination of the vulva revealed scarred external genitalia with dry skin. The glans clitoris, prepuce, right and left frenula, and the labia minora and majora were absent as shown in She was counseled for defibulation, to be performed under spinal anesthesia. A stab incision was made at the lower margin of the vulva and a Kelly clamp inserted under the scar tissue and a vertical incision made along the anterior surface of the infibulated scar until the original anatomical site of the clitoris was exposed superiorly and introitus inferiorly as shown in FGM/C prevalence in Ghana varies by region with a relatively high prevalence around 38% in Northern Ghana , 12, 14.In Africa, countries like Ghana and Senegal took initiatives by way of legislation to criminalize FGM/C. In 1994, an Act was enacted to amend the criminal code of Ghana, making FGM/C a criminal offense equivalent to second-degree felony liable to imprisonment of at least 3 years . HoweverIn the northern part of Ghana, it has been noted that FGM/C is common among daughters of women who have little or no form of education . AkweongThe patient in this report is a teenager who was subjected to infibulation as a minor as part of a sociocultural practice. She has lived with the consequences of this practice since it happened until the present moment. FGM/C is still being practiced in Ghana . Berg anThe pressing health problems of the patient we saw seem to have been resolved. However, a long-term follow-up would be needed to determine whether she may have other consequences, including potential psychological concerns .We reported a case of a patient experiencing sexual difficulties due to infibulation. Defibulation allowed this patient to engage in sexual intercourse with her partner within 6 weeks following surgery.


Background: non-hemorrhagic adrenal infarction (NHAI) is a rare cause of acute abdominal/flank pain during pregnancy; in order to ensure prompt and appropriate treatment, this diagnosis should not be overlooked. This case series highlights pertinent imaging findings, including ultrasounds (USs), computed tomography (CT), and magnetic resonance imaging (MRI) of recent NHAI cases. Methods: we compiled all consecutive NHAI cases from two university hospitals over a two-year period and checked the relevant clinical, laboratory, and imaging findings. Relevant articles on NHAI published from January 2010 to March 2021 were analyzed. Results: six cases were found in our database. CT-scans typically showed enlarged, hypodense, and non-enhanced adrenal glands. Unenhanced MRIs allowed for diagnoses and showed enlarged adrenal glands in the signal hyperintensity on T2 and diffusion-weighted imaging, without any signal hyperintensity on T1. In two of our six cases, USs showed swollen adrenal glands with fluid collection. Conclusion: NHAI and its differential diagnosis—in cases of acute pain during pregnancy—highlight the crucial roles of integrated radiological examination and cooperation between obstetricians and radiologists, both of whom should consider the location of the pain, the accessibility and tolerance of MRI, and the radiation exposure of CT. Despite its supposed poor sensitivity, an US performed because the patient reports pain should also be used to examine the adrenal gland regions. Non-enhanced MRI is clearly of value and access to it in emergencies is important to avoid radiation exposure Non-hemorrhagic adrenal infarction (NHAI) is a rare clinical cause of acute abdominal/flank pain during pregnancy. It should be differentiated from acute bilateral adrenal hemorrhage, which is often described in patients with hemostatic disorders. The clinical presentation of NHAI may vary from patient-to-patient, as well as the location of the pain . This could point to various common non-obstetric causes of acute pain, such as hydronephrosis, appendicitis, gallstones, and pulmonary embolism. Physicians and radiologists must agree on the need for imaging and define a challenging strategy. They should consider radiation exposure, diagnostic accuracy, and the availability of each imaging modality.If sonography is inconclusive, computed tomography (CT) is often performed as a second-line test despite radiation exposure, but magnetic resonance imaging (MRI) seems to be a good, non-irradiating alternative to establish the cause of pain . Among sThe purpose of this study, based on recent cases in two university hospitals, was to highlight the crucial role of imaging, including US, CT, and MRI. We also reviewed the previous cases reported in the literature.Between January 2018 and December 2020, 282 pregnant women were referred to the radiological departments of two university maternity hospitals for exploration of acute flank/upper abdominal/back–chest pain. Both hospitals have radiological databases where all reports are entered. We searched for adrenal gland anomalies among these reports and retrieved/confirmed NHAI in six women from ages 19 to 38 years (mean 29.5 years). All clinical and laboratory data and follow-up findings were retrospectively collected. All data analyzed were collected as part of routine diagnosis and treatment. The institutional review boards approved this study, and because of the retrospective nature of the study design, they waived the requirements for informed patient consent. All patients had abdominal and pelvic ultrasound examinations as first-line imaging. They also had contrast-enhanced CT. In two patients, CT pulmonary angiograms were required to rule out pulmonary embolisms, followed by abdominal CTs, because of the pathological findings at the lower parts of the chest CTs. Abdominal CTs were performed in two other patients because they had a history of kidney stones; two patients underwent dual energy contrast-enhanced CTs because of suspected appendicitis. Five patients had abdominal 1.5 T MRIs without gadolinium injection . All images were interpreted and reviewed by two radiologists with 7 and 31 years of experience in urological imaging. The diagnoses were defined as enlarged and hypoenhanced adrenal glands on the CT scans. Non-hemorrhagic components were defined by the absence of hyperdensity on non-enhanced CT scans and the lack of increased signal intensity in T1-weighted MR imaging. We searched the PubMed/MEDLINE databases for previous case reports using the keywords “non-hemorrhagic adrenal infarction”, “adrenal thrombosis”, and “pregnancy”.9/L; IQR 13.1–18.8) and increased C-reactive protein . Urine analysis and culture were normal for all women. An extensive search for a hereditary or acquired thrombophilic disorder proved negative, except for patient 6, who had a bilateral adrenal infarct (positive lupus anticoagulant without anti-cardiolipin or anti-beta-2 glycoprotein antibodies). This included homocysteine, proteins C and S, antithrombin III, factor II, VIII levels, lupus anticoagulant, anti-cardiolipin and anti-beta-2 glycoprotein antibodies, Leiden, prothrombin gene 20210A and JAK2 polymorphisms. Therapeutic dose anticoagulation was initiated for all patients. Patient 1 (37 weeks of gestation) had labor induction in order to manage analgesic treatments more easily. Patient 6 had a bilateral adrenal infarct; hydrocortisone supplementation started as soon as the diagnosis was confirmed by MRI. Under supplementation, plasma cortisol levels were near the lower limits of normal at 8 a.m. and 10 a.m. and the adrenocorticotropic hormone (ACTH) level was normal. She received an adapted treatment for the rest of her pregnancy and for delivery. Four months after her delivery, she still requires hydrocortisone supplementation. The other four patients had no further complications. Clinical, laboratory, and imaging findings are summarized in We diagnosed NHAI in six women . In all cases, NHAI occurred during the third trimester of pregnancy . None of the patients had a personal or family history of venous thromboembolism. Three had a history of kidney stones, including one during the first trimester. Pain was resistant with common analgesics, and opioid titration was required. Clinical and US pelvic examination findings were normal in all six patients and none were at risk of impending delivery. Laboratory tests showed leukocytosis  that we compiled, 16 involved the right adrenal gland. Only one involved the left adrenal gland and two patients had sequential and bilateral infarction. These two patients have subsequently developed adrenal insufficiency secondary to the infarction ,7. Pregnancy is a hypercoagulable state that predisposes the patient to thrombotic events. A pregnant woman has a relative risk of thrombosis five to six times higher than a non-pregnant woman of the same age . MoreoveFox et al. suggested, the central vein thrombosis appeared well organized; thus, it seems more probable that the central vein thrombosis had resulted in sinusoidal thrombosis [. The location of the pain may vary from patient-to-patient, including back–chest, abdominal, and flank pain. This induces various supposed etiologies, including pulmonary embolism, renal/biliary colic, pyelonephritis, and appendicitis. Therefore, radiologists should be aware of this rare diagnosis and its variable clinical expression. Because of differential diagnoses, especially hydronephrosis, US screenings should remain as first-line imaging modalities, but in adults, it does not allow the diagnosis of NHAI with acceptable accuracy. Nevertheless, the unusual visualization of one adrenal gland should suggest its swelling, as in two of our cases. Because of its efficiency and safety, non-enhanced MRI should be the preferred second-line modality for pregnant women [The pathophysiology of NHAI remains unclear, but involves thrombosis of the adrenal vena. The histological analysis of adrenal infarction strongly suggest that the thrombosis of the main adrenal vein is the cause of the infarction. As rombosis . A possirombosis , appearsrombosis . The typrombosis . The locnt women . It typint women . Just asnt women . Our imaMRI may help to differentiate physiological ureteral dilatation of pregnant women from pathological dilatation, with peripheral renal edema and renal enlargement. However, urinary stones can be challenging to identify on MRIs. CTs allow for specific diagnoses of renal colic due to ureteric stones and the functional consequences, which are characterized by a delayed nephrogram . The latThe main limitation of our study is the small sample of patients, despite the multicentric recruitment. This limitation is inherent to the rarity of the pathology. We are aware of the operator-dependent nature of ultrasonography, nevertheless, the adrenal area anomalies highlighted in our study were seen by two different sonographers. This should encourage practitioners to explore the adrenal aera, especially since modern ultrasound scans allow exploration of anatomic regions—areas that were previously supposed to be inaccessible.For acute flank/abdominal pain during pregnancy, we suggest simplified imaging management. After an US, unenhanced abdominal and pelvic MRI should play an essential role in the etiological investigation of acute abdominal pain in pregnant women. If pulmonary embolism is initially suspected, the CT pulmonary angiogram remains essential, and radiologists should pay special attention to the adrenal glands, which are in the field of view. Abdominal/pelvic CTs should be performed if the MRI is inconclusive. Regarding our case series, abdominal CTs could have been avoided in every case if the MRI had been the second radiological exam after the US. Imaging plays a central role in the management of abdominal pain during pregnancy. The clinical presentation of NHAI is non-specific, but imaging findings are typical and highly similar from patient-to-patient.US examination performed for abdominal/flank pain should screen the adrenal gland region.Our results highlight the usefulness of unenhanced MRIs, which should include diffusion-weighted imaging.A prompt diagnosis of NHAI induced the implementation of anticoagulation and screening for acute adrenal insufficiency.


Inflammatory myofibroblastic tumour (IMT), also known as plasma cell granuloma (PCG) or inflammatory pseudotumour (IPT), is a distinctive, rarely metastasizing neoplasm composed of myofibroblastic and fibroblastic spindle cells accompanied by inflammatory infiltration of plasma cells, lymphocytes and/or eosinophils. IMT predominantly affects children and young adults, and the age at presentation ranges from 3 to 89 years. We present a very rare case of recurrent testicular IMT without ALK rearrangement. This case highlights the clinical characteristics and diagnostic factors associated with primary and recurrent foci of this rare tumour, along with key therapeutic approaches. Inflammatory myofibroblastic tumour (IMT), also known as plasma cell granuloma (PCG) or inflammatory pseudotumour (IPT), is a distinctive, rarely metastasizing neoplasm composed of myofibroblastic and fibroblastic spindle cells accompanied by inflammatory infiltration of plasma cells, lymphocytes and/or eosinophils , 2. IMT The patient was a 58-year-old man who was admitted to our hospital for mass in the left scrotum that had persisted for over 6 months, accompanied by scrotal pain and discomfort and frequent, urgent and incomplete urination. Nocturia occurred 3–4 times per night without painful urination. The patient did not experience gross haematuria, fever, back and abdominal pain, loss of appetite or weight loss. Diagnosis and treatment of the patient is shown in Routine blood test results, blood biochemistry analyses, tumour marker levels and hormone levels  were normal. CT of the pelvic cavity showed a heterogeneous mass in the left scrotum, which was considered malignant because the mass is obviously enhanced in the arterial phase Figure 2Informed consent was obtained from the patient to perform left groin exploration with left radical orchiectomy. A round, smooth and hard mass with a clear boundary measuring approximately 10×8 cm was observed during the surgery. The left spermatic cord was ligated and the testis was removed. The postoperative pathological examination revealed abnormal testis parenchymal structure with large numbers of infiltrating lymphocytes and plasma cells, lymphatic follicles, and proliferating fibroblasts and myofibroblasts; additionally, scattered R-S cells could be seen in some areas Figure 3th month of follow-up due to the slow and painless growth of the mass in the left lower abdominal wall. A physical examination revealed that the mass was immobile, approximately 4×3 cm in size and tough in quality. B-ultrasonography indicated a hypoechoic solid mass in the proximal area of the left lower abdominal wall, approximately 4.3×3.1×2.5 cm in size. The boundary was unclear and irregular, and the echo was not uniform. CDFI revealed low blood-flow signals. Enhanced pelvic CT examination also showed the same result; thus, the tumour was considered to be in situ IMT , human chorionic gonadotropin (hCG), lactate dehydrogenase (LDH), and placental alkaline phosphatase (PLAP) are generally and significantly elevated in patients with testicular germ cell tumours . CorrespB-ultrasound, CT, and magnetic resonance imaging are commonly used imaging examinations to diagnose scrotal masses. Although imaging examinations play an important role in diagnosing IMT, postoperative histological examination is the gold standard , 24. TheThe definitive diagnosis of IMT is dependent on postoperative pathological and immunohistochemical examinations. Histologically, IMT is characterized by varying proliferation of spindle cells in a myxoid to a collagenous matrix, and a significant inflammatory infiltrate consisting mainly of plasma cells and lymphocytes with occasional eosinophils and neutrophil involvement . Three tImmunohistochemically, 90% of IMTs express SMA and 50% express pancytokeratin focally. IMT is typically positive for low-molecular-weight cytokeratin and negative for high molecular weight cytokeratin P63 and epithelial membrane antigen. Approximately 50% of IMTs are positive for desmin . Approxiin situ relapse can occur despite extended dissection  for the publication of any potentially identifiable images or data included in this article.JL was involved in the identification, selection, and management of patient cases and wrote and revised the manuscript. SL, ZB, SZ, CL, and QL was involved in the management of patient cases. QL finally reviewed the manuscript. All authors contributed to the article and approved the submitted version.Supported by Tianjin Key Medical Discipline Construction Project; Science and Technology Fund of Tianjin Health and Family Planning Commission (No. 16KG103); Tianjin Health Science and Technology Project (No. ZC20162).The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Vascular malformations of the maxillofacial region are unusual, and they occur more rarely in bone than in soft tissue. Mandibular intraosseous vascular lesions represent 0.5-1.0% of all bone tumors, and they are classified as venous malformation, lymphatic malformation, arterial malformation, arteriovenous malformations, and arteriovenous fistulae. Venous malformation is the most common vascular malformation, accounting for 44-64% of all vascular malformations, and is considered a low-flow malformation. Endovascular therapy as selective angiographic embolization is considered as the first-choice treatment associated or not with emboli injections with a success rate of 70%, and this evades mutilating surgery and related sequelae. We report a case of mandibular venous malformation on a 45-year-old female complaining of unilateral swelling of the left body of the mandible with facial deformation. The computed tomography scan images and the T1-weighted MR images showed a lesion that expresses an expansible lesion in the spongy bone of the left of the mandible with a buccal cortical rupture. Signal voids were not identified, suggesting a low-flow vascular lesion. The T2-weighted images exposed hypersignals; accordingly, a vascular lesion was suspected. The treatment was done under locoregional analgesia; after selective angiography, direct histoacryl injection was completed, followed by bone cement injection. The patient was followed yearly since1998. Radiological images of 10-year follow-up MRI showed a stabilization of the lesion without any new extensions. The panoramic radiograph after 22 years showed a bone formation inside the body of the mandible. The long follow-up period and the absence of any complications are favorable for the adopted treatment plan. Vascular lesions were considered as hemangiomas before the1980s, and it was in 1982 that Mulliken and Glowacki proposed a classification for vascular anomalies based on pathological features [VMs include capillary malformation (CM), venous malformation (VM) , lymphatic malformation (LM), arterial malformation (AM), arteriovenous malformations (AVM), and arteriovenous fistulae (AVF) .In addition, VMs can be divided based on the degree of blood flow: “slow flow” , “fast flow” , and combined vascular malformations .VMs are the most common vascular malformation, accounting for 44-64% of all vascular malformations, and are considered as low-flow malformations [Management of VMs is very complex and needs a multidisciplinary team for successful results . At presWe describe a case of intraosseous multilocular VM of the mandible where a conservative treatment was applied with a follow-up period of 22 years.A 45-year-old female was oriented to our department complaining of unilateral swelling of the left body of the mandible with facial deformation.Clinical exam revealed a well-delimited hard swelling at the left of the mandible (body and angle) with submandibular adenopathy. Intraorally, a hard tumefaction that fills the buccal posterior area of the mandible was noticed. Mobility scale 3 of the second premolar was observed, and no paresthesia was noted Figures .History reveals that the swelling started three years ago without any pain, and the left mandibular first and second molars were extracted as they were mobile without any complications or hemorrhage.A 2D-panoramic radiograph showed then a well-defined multilocular radiolucency, spreading from the left mandibular mental nerve region anteriorly to the ramus posteriorly; small loculations and fine trabeculae were noted inside the lesion .μL).Blood test analysis was performed, and the results were in the range of normality  showed a single layer of endothelial cells lining the dilated vascular spaces with mild inflammatory polymorphous leucocytic infiltrates in the surrounding tissue with thin-walled and dilated vascular spaces of variable diameter Figures .Following the clinical, radiological, and histological results, a CT scan and MRI were requested to determine the soft and hard tissue extensions of the lesions.The CT scan images presented an expansible multilocular lesion with a distinct periosteum reaction on the buccal cortical bone. No loculations were detected, and the axial images displayed a trabeculated lesion in the body of the left mandible .MRI images with 1.5 T using routine T1- and T2-weighted spin-echo sequences showed the existence of a lesion of the entire left part of the body of the mandible and extending to the ramus without reaching the condyle.The T1-weighted MR images showed a lesion with hyposignals that express an expansible lesion in the spongy bone of the left of the mandible with a buccal cortical rupture. Signal voids were not identified, suggesting a low-flow vascular lesion .The T2-weighted images exposed hypersignals; accordingly, a vascular lesion was suspected. Axial cuts displayed high signal intensity and an expansible lesion in the bone marrow of the left hemimandible, with a buccal bone discontinuity and extension towards the soft parts of the vestibular edge of the mandible, producing a subcutaneous tumefaction. Inner loculations were present in the axial cuts suggesting multilocularity .Clinical, radiological, and histological findings leaded to a conservative treatment, based on embolization and bone cement injections. The treatment plan was suggested and discussed with the patient and her family who signed a consent form.Angiography, through a transfemoral approach, was achieved under local anesthesia in the hospital, and selective arteriography of the left external carotid artery was performed.Digital subtraction angiograms clearly displayed the facial and its branches, inferior alveolar, and left lingual arteries .After this and under locoregional analgesia of the left mandible, direct histoacryl injection was completed with a low speed under fluoroscopy monitoring, in the angiographic suite through a percutaneous puncture performed with a 22-gauge puncture needle.The volume of the histoacryl used varied around 4 to 5 ml (diluted 25% with lipiodol) Figures .Then, bone cement with gentamicin (CMW3™) mixed with lipiodol was injected through the same 22-gauge puncture needle, first in the posterior part of the lesion , followeThe percutaneous injections revealed abnormal venous lakes or pouch which depicts the venous lesion.No complications were noted in the postoperative period, and the patient was discharged the same day. The patient was followed yearly from 1998 till 2020 Figures .The swelling was maintained with the same volume, and the only difficulties were losing her mandibular teeth.Radiological images of 10-year follow-up MRI showed a stabilization of the lesion without any new extensions Figures .VMs are characterized by the irregular proliferation of endothelial cells of the blood vessels. They are considered as congenital inaccuracies of vascular morphogenesis that result in fragile endothelial cell proliferation inside the network of blood vessels .VMs appear more frequently in soft tissue than in bone. In the mandible, they are very rare and represent only 0.2–1.0% of all intraosseous tumors, and the most common symptoms are pain, swelling, and tooth mobilities , 11. PreSurgery is considered as the last treatment choice for AVMs of the mandible , 5. ReseEndovascular therapy has been proposed for the treatment of the mandibular VMs with a success rate of 70%, and this evades mutilating surgery and related sequelae , 5. AddiInjection of emboli is suggested in a direct intraosseous technique; embolization should be achieved before to reduce the blood flow and to admit more precise accumulation of bone cement inside the lesion .The biomaterials used as injectable embolic for the treatment of vascular lesions are intended to become occlusive by provoking thrombosis or directly obstructing the lesion when hardening of the embolic appears .Different biomaterials have been used such as polymethylmethacrylate, n-butyl-2-cyanoacrylate, cellulose acetate polymer, fibrin glue, and bone cement .In general, intraosseous VMs display hyposignals on T1WI and hypersignals on T2WI . Slow-flThe present case displayed homogenous hyposignals on T1WI and hypersignals on T2WI. The absence of signal voids advocates that we are facing a vascular malformation classified as a slow-flow lesion.The MR images showed that the lesion was multilocular with bone cortical discontinuity with extension to the soft tissue.The conservative treatment adopted a superselective angiographic embolization with the direct injection of histoacryl mixed with lipiodol and followed with the injection of bone cement in the two parts of the lesion. The first step reduces blood flow, and the second step is aimed at inducing thrombosis and at directly blocking the lesion.The MR images 10 years later revealed stabilization of the size and extensions of the lesion. The panoramic radiograph after 22 years showed a bone formation inside the body of the mandible. The long follow-up period and the absence of any complications are favorable for the adopted treatment plan.VMs need a very close multidisciplinary collaboration involving interventional radiology, otorhinolaryngology, oral/maxillofacial surgery, and plastic surgery in the arrangement and application of an appropriate treatment plan for these lesions.


Left paraduodenal hernia (PDH), may present as a surgical emergency with an increased risk of strangulation and incarceration. The diagnosis is challenging because of the non-specific presentation. In the absence of common epigastric or upper abdominal pathology and non-resolving symptoms, a high index of suspicion is required to diagnose left PDH. This report describes a case of radiologically diagnosed left paraduodenal hernia and subsequent successful surgery. It also includes a review of midgut embryology, and the anatomy and radiology of left PDH. Hence, this entity may present a clinical challenge and imaging, especially if symptomatic, is imperative. Paraduodenal hernias (PDH) are the commonest of the internal hernias and are classified into left and right, with the former being more prevalent.1Abdominal hernias are categorised into external and internal hernias. External hernia represents herniation of intestinal loops through a defect in the wall of the abdomen or pelvis. An internal hernia is defined by herniation of a viscus through a normal or abnormal peritoneal or mesenteric aperture within the peritoneal cavity. Internal hernias are rarely encountered in general practice, however, on the background of increased liver transplantations and gastric bypass surgery for bariatric treatment, the incidence of internal hernia is becoming more prevalent.The index patient was a 57-year-old male who presented with vague, intermittent, recurrent abdominal pain. The pain evolved into a worsening epigastric pain radiating to the back with associated vomiting. The supine plain radiograph of the abdomen  demonstrGiven the presence of a left upper quadrant mass on plain radiography that was suboptimally appreciated on ultrasound, computed tomography (CT) was requested. Multiphase contrasted CT imaging was performed with prior administration of oral contrast . A clustIntraoperative findings  confirmeThis article followed all ethical standards for research.Consent was acquired from the patient to include the data and images in the manuscript. Data and images in the manuscript were anonymised.1Left paraduodenal hernia represents an internal abdominal hernia. Internal hernias are defined by the protrusion of a viscus through a normal or abnormal peritoneal or mesenteric aperture within the peritoneal cavity. The orifice may be congenital or acquired, secondary to post-surgical, post-inflammatory or a traumatic defect. Congenital defects include normal apertures, such as foramen of Winslow, and abnormal apertures arising from anomalies of internal rotation and peritoneal attachments.In understanding the pathogenesis and clinical manifestations of left PDH, it would be imperative to review the embryology of the normal sequence of events relating to the midgut position. The midgut is suspended in the midline by its dorsal mesentery and undergoes a sequential pattern of rotations that is divided into three stages .2 who reported the first stage beginning at the 5th week in the life of the embryo, in which rapid growth of the abdominal viscera forces a greater component of the midgut external to the abdominal cavity.This was well described by Bartlett et al.In the 10th week of the life of the embryo, the abdominal cavity increases in size and the midgut gradually returns into the abdominal cavity, beginning the second stage of rotations. The midgut has now rotated 90 degrees in a counterclockwise direction on the axis of the superior mesenteric artery, with the pre-arterial segment occupying the right side and the post-arterial segment occupying the left side of the abdominal cavity. The pre-arterial segment rotates an additional 180 degrees counterclockwise, initially posteriorly and subsequently to the left of the superior mesenteric artery, coming to lie to the left of the midline in the abdominal cavity. The post-arterial segment rotates, led by the caecum, which passes counterclockwise, anterior to the superior mesenteric artery into the right upper quadrant of the abdomen, occurring between the 10th and 11th week. The caecum may not completely descend into the right lower quadrant until the end of the 5th month in the life of the embryo.The third stage represents the fusion of mesenteries and fixation of the midgut. A leaf of the mesentery of the colonic portion of the midgut that has a posterior position, fuses with the peritoneum of the posterior abdominal wall and the space is obliterated. The mesentery of the small bowel is fixed to the posterior abdominal wall and mesentery of the duodenum fuses with the posterior parietal peritoneum.4 Abnormalities in the rotation of the pre-arterial segment in the second stage as it rotates posteriorly and then to the left of the superior mesenteric artery results in a left PDH. During this process, the bowel invaginates into an unsupported area of the descending mesocolon resulting in the anterior margin being formed by the ascending branch of the left colic artery and the inferior mesenteric vein. The small bowel comes to lie in a sac, lined by peritoneum, posterior to the mesentery of the descending colon. The terminal ileum enters the peritoneal cavity through the neck of the sac to reach the caecum  the presence of a fossa (fossa of Landzert), (2) the presence of the inferior mesenteric vein in the neck of the sac and (3) sufficient mobility of the small bowel to allow it into this sac derived from this fossa.The fossa of Landzert is located to the left of the fourth part of the duodenum, extending posteriorly to the descending mesocolon. The opening is immediately inferior to the duodenojejunal junction and bordered anteriorly by the inferior mesenteric vein and ascending branch of the left colic artery.7 Left PDH is three times more common than right PDH with a male predilection of 3:1.7 Left PDHs usually present in the fourth to sixth decade, with a mean age of presentation at 38.5 years.7Paraduodenal hernias are the most common type of internal hernias, accounting for 50% of cases.8 Patients commonly present with postprandial pain typically chronic in nature and dating back to childhood.1Paraduodenal hernias often present a clinical challenge. Presentation may range from acute intestinal obstruction, which is the most common presentation, to vague abdominal pain, often relieved by changes in position.8 Our patient demonstrated a well-circumscribed ‘mass’ projected in the left upper quadrant of the abdomen, with preservation of the left psoas outline and no left lower rib lesions.Plain radiographs may demonstrate a circumscribed ovoid mass of jejunal loops occupying the left upper quadrant of the abdomen immediately lateral to the fourth component of the duodenum.9The associated risk of strangulation and intestinal infarction of more than 50% over the course of a lifetime, makes it necessary to investigate symptomatic patients. The high rate of mortality associated with these complications justifies the role of CT early in the pre-operative diagnosis of PDH.10 The ascending branch of the left colic artery and the inferior mesenteric vein form an important CT landmark along the anterior margin of the hernia.The characteristic CT appearance consists of an abnormal cluster or sac-like mass of dilated small bowel loops lying between the stomach and pancreas, to the left of ligament of Treitz. There is usually mass effect that displaces the posterior wall of the stomach, the duodenal flexure inferiorly and the transverse colon inferiorly. The mesenteric vessels supplying the herniated small bowel, appear crowded, engorged and stretched at the entrance of the hernial sac.Our patient presented with vague, intermittent and recurrent abdominal pain that was progressively worsening. The impression of a mass within the left upper quadrant of the abdomen on plain radiography was characterised at CT, which demonstrated pathognomonic features of a left PDH. Urgent surgical intervention confirmed the diagnosis and the patient recovered without complications.11This case report aimed to highlight the importance of plain radiograph interpretation in the background of an internal hernia, which if present, will allude to underlying pathology. Although this may be non-specific, it will support the need for further cross-sectional imaging to delineate the pathology. The case report further describes and diagrammatically illustrates the pertinent aspects of midgut embryology, allowing the radiologist to appreciate the pathogenesis of a left PDH. Identifying the abnormal configuration of the proximal small bowel and associated vascular landmarks assists in the correct interpretation and diagnosis. Internal hernias are rarely seen in clinical practice and it is important for radiologists to familiarise themselves with this concept, as the adverse outcome of an unsuspected or unidentified PDH can result in ischaemia, strangulation and obstruction with mortality between 20% and 50% due to delayed management.Left PDH represents a clinically challenging and elusive diagnosis. An understanding of the mesenteric, peritoneal folds, anatomy of the duodenal fossa and embryology of the midgut is important to make the diagnosis. It is imperative for radiologists and surgeons to familiarise themselves with this uncommon condition, especially in patients presenting with non-resolving vague epigastric pain. Plain radiograph may be suggestive, however, CT remains the gold standard for the diagnosis.


Background: Pleomorphic liposarcoma (PLS) is a very rare type of primary pulmonary sarcoma. Clinical data about these tumors are limited, and optimal treatment has not yet been defined.Case Report: A 32-year-old male presented with pain and heaviness over the right chest. Contrast-enhanced computed tomography (CECT) of the chest showed a heterogeneous hypodense pleural-based mass and a mediastinal mass. The patient was treated with chemoradiotherapy, followed by excision and adjuvant chemotherapy. Seven months after completion of treatment, he presented with an abdominopelvic mass and soft tissue peritoneal deposits. The mass was resected, and second line chemotherapy resulted in a partial response. The patient was routinely followed. Six months after completion of the second round of chemotherapy, CECT showed multiple soft tissue deposits in the right lumbar region, right hemipelvis, and presacral region with no evidence of pulmonary disease. Chemotherapy elicited a partial response. Three years from the date of diagnosis, the patient was alive with stable disease.Conclusion: This case is unique because of the rare primary site of PLS presentation and the rare presentation of peritoneal metastasis. Citing such cases would help us to define adequate treatment protocols for this aggressive tumor. PLS is rare, accounting for only 5% to 10% of all lipomatous tumors.2 PLS is characterized by a high rate of local invasion, metastasis, and recurrence with an aggressive clinical course and is therefore considered to be the most malignant of the liposarcomas.3 To the best of our knowledge, only 15 cases of primary pulmonary liposarcoma had been reported as of December 2020.14 Of these 15 cases, only 4 patients had PLS histology.14Liposarcoma is a relatively common soft tissue sarcoma, but primary pulmonary liposarcoma is a rare neoplasm. Primary pulmonary sarcomas are extremely rare and account for <1% of all lung tumors.We present a case of primary pulmonary PLS with a rare pattern of peritoneal metastasis that was managed with multimodal treatment.A 32-year-old male presented in March 2017 with complaints of pain and heaviness over the right chest for 2 months, aggravated by coughing and deep inspiration. The patient also reported shortness of breath on exertion. He was a chronic smoker (5 pack-years) and binged on alcohol on occasion. Clinically, breath sounds were absent over the right basal zone. Chest x-ray revealed homogenous opacity involving the right hemithorax. Baseline contrast-enhanced computed tomography (CECT) of the chest revealed a well-defined 20.8 × 13.6 × 13-cm pleural-based heterogeneous hypodense mass (10-12 Hounsfield units) along the right lateral chest wall, medially extending into the mediastinum and compressing the heart with collapse of the right lung A. Bronch2, maximum dose capped at 2 mg)/doxorubicin (75 mg/m2)/cyclophosphamide  every 3 weeks. Response assessment after 3 cycles via CECT revealed no substantial change in tumor size, and the patient then underwent hypofractionated radiotherapy to the lung mass with a dose of 30 Gy in 10 fractions for 2 weeks, with 6 MV photons using parallel-opposed fields.The diagnosis of primary PLS of the lung was made after PET-CT and the extensive immunohistochemistry panel on the biopsy specimen ruled out the possibility of other primary sites. The tumor was deemed inoperable because of its large size and the disease extension, so the patient was considered for chemoradiotherapy. To downsize the tumor, the patient was started with a regimen of vincristine /etoposide (100 mg/m2) every 3 weeks. The patient tolerated the therapy well, and posttreatment CECT at 6 weeks revealed no residual disease. The patient was followed up with clinical examinations every 2 months, and CECT at 3 months did not show any evidence of disease.A partial response was noted after radiotherapy, and the patient underwent excision of the tumor via the median sternotomy and anterolateral thoracotomy approach. Postoperative histopathology showed PLS with focal myxoid changes and large areas of necrosis. A specimen of the diaphragm showed tumor infiltration. After surgery, the patient received 6 cycles of ifosfamide , and cystic (hemorrhagic) with capsule rupture, and it was removed in pieces. The histopathology of both the thoracic and abdominal tumors showed features of PLS A and 2B 2 on days 1 and 8)/docetaxel (75 mg/m2) every 3 weeks. Interval response evaluation according to the Response Evaluation Criteria in Solid Tumors (RECIST) showed partial response, and chemotherapy was continued for 3 more cycles. Imaging after treatment completion showed stable disease in the abdomen with peritoneal deposit. Because the patient was asymptomatic, no further active treatment was planned, and the patient was followed with clinical examinations every 2 months and radiologic evaluations every 3 months.The patient was treated with 3 cycles of gemcitabine  every 4 weeks. Two months after completion of chemotherapy, CECT showed partial response.Six months after completion of the second round of chemotherapy, the patient presented with intermittent pain over the right iliac fossa. CECT showed multiple soft tissue deposits in the right lumbar region, right hemipelvis, and presacral region with no evidence of pulmonary disease . The patThree years from the date of diagnosis, the patient was alive with stable disease. During the treatment period, he received 4 cycles of chemotherapy.4 The earliest reported case of primary pulmonary liposarcoma was in the 1940s by Latienda and Itoiz.5 Liposarcomas originate from primitive mesenchymal cells and are common in the extremities or retroperitoneum. For primary intrathoracic liposarcoma, the mediastinum is the most common site of origin; primary liposarcomas rarely arise from lung parenchyma, chest wall, or pleura.3 Hochberg and Crastnopol analyzed 77 patients with primary pulmonary sarcoma, and the most common variants observed were fibrosarcoma and leiomyosarcoma, with liposarcoma seen in only 2 patients.6 The Sarcomas constitute <1% of all pulmonary malignancies, and PLS is the least common variant.13 However, in our case, no such risk factors were identified. Primary pulmonary liposarcomas present with symptoms similar to those of lung cancer, such as cough, sputum production, hemoptysis, dyspnea, chest pain, and loss of weight and appetite. Because of similar symptomatology, primary pulmonary liposarcoma may be misdiagnosed as pulmonary tuberculosis in developing countries such as India because of the high prevalence of that disease.8 Approximately 10% of all primary pulmonary liposarcomas are asymptomatic, which may be because of the expansile nature of the tumor rather than infiltration.6 Also, because these tumors are slow growing, they often present as a large mass. The absence of any characteristic symptom further delays diagnosis.Based on published cases, primary pulmonary liposarcomas occur in patients with a wide age range (9 to 61 years) and a median age of 46.5 years, present earlier than lung cancer, and have no sex predilection. Our patient presented at 32 years of age. Possible risk factors associated with development of primary pulmonary liposarcoma are pleuropulmonary asbestosis or malignant transformation of pulmonary lipoma.9 In this case, we observed extrinsic compression of the tracheobronchial tree, but no exophytic mass could be appreciated on bronchoscopy.Primary pulmonary liposarcoma can primarily involve the lung but can also be completely pleural in origin, such that in advanced cases, distinguishing a pulmonary from a pleural mass can be difficult. Usually, these tumors involve lung parenchyma and therefore are not visualized during bronchoscopy. Only 2 cases of primary pulmonary liposarcoma have been reported with positive findings on bronchoscopy, seen as polypoid masses and causing near total obstruction.10 Also, on magnetic resonance imaging, the mass showed high signal intensity on T2-weighted images and low intensity with fat-saturated T2-weighted images. In our case, CT showed similar findings. Therefore, the differential diagnosis of primary pulmonary liposarcoma is possible from imaging. Peritoneal dissemination has been cited only once in a case of undifferentiated pleomorphic sarcoma.15 To the best of our knowledge, the extensive dissemination seen in our case has not been reported before in a case of primary pulmonary PLS.Said et al described the radiologic findings of primary pulmonary liposarcoma: a well-defined lobulated inhomogeneous hypodense mass interspersed with multiple areas of very low densities, suggestive of fat.3 On immunohistochemistry, well-differentiated, myxoid/round-cell, and dedifferentiated liposarcomas are positive for vimentin, S-100, MDM2, and CDK4. PLS is usually negative for MDM2 and CDK4 but expresses p53. PLS resembles a malignant fibrous histiocytosis-like tumor, characterized by the predominance of pleomorphic spindle-shaped cells with occasional giant multinucleated cells.16 Fletcher emphasized the difficulty in distinguishing PLS from other high-grade pleomorphic sarcomas in his review of 159 such tumors; 21% were reclassified as PLS on review.17Liposarcomas are categorized into 4 broad histologic groups. Well-differentiated liposarcomas  have a better prognosis compared to myxoid/round cell–variant (intermediate prognosis), dedifferentiated, and pleomorphic tumors, which have poor prognoses as per the 2012 National Comprehensive Cancer Network classification.3 They observed that tumor size, distinct tumor locations, surgery, age at presentation, and sex had no impact on overall survival (OS) and disease-free survival (DFS). Radical surgery had a significant effect on OS, and histologic subtype also had significant impact on both OS and DFS.3 However, when compared with other primary sites, pulmonary primaries had a worse prognosis. Despite negative surgical margins, early disease dissemination has been observed, probably because of microrupture or frank rupture observed during dissection of cystic masses. Disseminated disease was seen in our patient as well, with capsule rupture and the tumor having to be removed in pieces, which may have led to peritoneal seeding.Chen et al studied various prognostic factors for primary intrathoracic liposarcoma.14 Chen et al analyzed 23 cases of primary intrathoracic liposarcoma and observed that patients having radical surgery had significantly better OS (P=0.029) than those without surgery.3 They also reported that metastasis was frequently seen, predominantly in the lung, pleura, liver, and bone, most commonly of the dedifferentiated tumor type.Treatment options for primary pulmonary liposarcoma are based on the algorithms used for management of primary liposarcomas in the extremities and retroperitoneum. Radical excision with lymph node dissection is the preferred treatment modality, and best survival is reported with R0 resection.18Chemotherapy and radiotherapy as adjuvant treatment in those with residual disease, inoperable cases, or metastasis have shown limited success in soft-tissue sarcomas. Doxorubicin when given alone or in combination with ifosfamide results in significant response in sarcomas.19 In sarcomas of the extremities, adjuvant radiotherapy after limb-sparing surgery improved local control. This improved control with adjuvant radiotherapy can be extrapolated to pulmonary liposarcomas. Myxoid liposarcomas are radiosensitive, and excellent responses have been observed with neoadjuvant radiotherapy.20 However, radiotherapy improves only local control with no improvement in survival.19In 2016, Nassif et al discussed advances in the management of liposarcomas—predominantly primaries of the extremities and retroperitoneum—but the same management can be tried in pulmonary primaries.22 The US Food and Drug Administration has approved trabectedin  and eribulin  as second-line agents for treatment of liposarcoma.25Liposarcomas of the extremities show good response to systemic chemotherapy. For metastatic disease, the combination of ifosfamide/doxorubicin or gemcitabine/docetaxel results in a 16% to 43% response and OS of 18 to 30 months.Primary pulmonary PLS is an extremely rare neoplasm. This case is unique because of the rare primary site of PLS presentation and the rare presentation of peritoneal metastasis. PLS behaves as a high-grade tumor with frequent metastasis. Because limited data are available, standard treatment guidelines have not been formulated, and treatment is principally extrapolated from experience in treating liposarcomas of the extremities or retroperitoneum. Reporting more cases would help us understand the natural behavior of these aggressive malignancies to optimize treatment options and follow-up.


The objective is to describe a rare case of lumbar lipomyelomeningocele presenting as progressive urinary incontinence. Lipomyelomeningocele is a type of closed spinal dysraphism typically presenting as a lipomatous mass contiguous with a neural defect above the gluteal crease. Tethered cord syndrome is defined as symptoms and signs caused by excessive spinal cord tension from an abnormally low conus medullaris, with an abnormally thick filum terminale attached to the lower sacral region. A 19-year-old male with no remarkable medical history presented with low back pain and urinary incontinence for the past one year. On physical exam patient had normal motor strength, sensory testing to all modalities was intact. The rectal tone was normal, and no saddle anesthesia was noted. MRI lumbar spine revealed lumbar lipomyelomeningocele with associated tethered cord syndrome. The patient underwent tethered cord release surgery with lipoma excision. Pathology of the soft tissue showed fibrovascular tissue and mature adipose tissue consistent with lipoma. The majority of cases of tethered cord syndrome are related to spinal dysraphism, a rare pediatric syndrome. It is potentially treatable if caught early, and MRI can help with an accurate diagnosis of the condition. Older adults are more likely to present with urological and neurological complaints. Surgical un-tethering is indicated in patients with progressive symptoms. In our case, the only presenting symptom was urinary incontinence, and the neurological exam was normal other than lower lumbar paraspinal tenderness. Lipomyelomeningocele is a rare congenital type of closed spinal dysraphism that typically presents as a lipomatous mass contiguous with a neural defect. It is usually located in the lumbar region above the gluteal crease and is found in 0.3 and 0.6 per 10,000 live births . Unlike A 19-year-old right-handed Hispanic Spanish-speaking male with no prior past medical history presented to the hospital with lower back pain, urinary incontinence for one year. He has been wearing diapers daily. He denied any motor weakness or paresthesias in extremities, bowel incontinence, or saddle anesthesia. On physical exam his mental status assessment and cranial nerve examination were normal. The sensation was intact to all modalities throughout, and motor strength was normal. Deep tendon reflexes were 2+ throughout and no pathological reflexes were noted. The rectal tone was intact and there was no saddle anesthesia. He did have a circular bump in his lumbar region measuring approximately 10 cm x 10 cm, with no skin dimple or tuft of hair. In the emergency department, over 1,200 mL of residual urine was removed through a straight catheter. MRI of the lumbar spine revealed a low-lying conus, thickened filum, and a fibrolipoma inserting into a terminal lipoma continuous with subcutaneous fat through a defect in the sacrum Figures , 1B. He Lipomyelomeningocele is a form of closed neural tube defect and it can present with neurological deficits secondary to a tethered cord . The uriOur patient did undergo surgical un-tethering, although the surgery, unfortunately, did not improve the patient’s symptoms. Patients who primarily have motor or sensory deficits are more likely to improve following surgery compared to those with urinary or sexual dysfunction. Urological dysfunction appears to be more reversible in younger patients, especially infants . ComplicIn this report, we discussed a rare case of lipomyelomeningocele presenting in adulthood with a one-year history of urinary incontinence. No other neurological symptoms were present, and the neurological exam was normal aside from lower lumbar paraspinal tenderness. MRI proved essential in making the diagnosis. This case highlights the importance of MRI in detecting the condition, and it should be noted that early diagnosis is important for improving outcomes of surgical treatment. Early surgery is indicated to prevent cord infarction, paraplegia, and bowel and bladder dysfunction.


Intracranial mature cystic teratoma is rare. The temporal lobe is a very unusual location and can pose a considerable diagnostic challenge. Proper histological diagnosis and long‐term follow‐up are imperative. Intracranial mature cystic teratoma is rare. The temporal lobe is a very unusual location and can pose a considerable diagnostic challenge. Proper histological diagnosis and long‐term follow‐up are imperative. Before the onset of an episode, he had an aura in the form of blurring of affect for a brief period, followed by multiple episodes of abnormal movements of the lips and tongue lasting for about 1 min which he was unaware of, with each episode lasting for approximately 4–5 s. He did not have generalized body movements, uprolling of the eyes, clenching of teeth, tongue bite, frothing from the mouth, and urinary/fecal incontinence during the attack. Following the episode, he experienced postictal confusion and headache for a few minutes. These seizure episodes had resulted in minor accidents, falls, and trauma. The frequency of these episodes gradually increased from once a month initially to 4–6 times a day in the past few months. There was no history of loss of consciousness and neurological deficits, limb weakness, visual diminution, fever, headache, weight loss, vomiting, or features suggestive of raised ICP.On neurological examination, the Glasgow coma scale was 15/15 with regular reactive pupils and normal fundoscopy. All the cranial nerves were intact. Motor power was 5/5 (MRC grade) across all major joints with an intact sensory examination. All other systemic examinations were unremarkable.Magnetic Resonance Imaging (MRI) of the brain revealed approximately 5.8 cm × 5.5 cm × 5.2 cm sized well‐defined T1 heterogeneously low, T2 heterogeneously high‐signal intensity cystic mass with thin septations in the right temporal lobe with mild patchy restriction of diffusion in diffusion‐weighted imaging (DWI) image without any solid component in it. The lesion showed peripheral wall enhancement in post‐contrast images alongside mass effect causing effacement of adjacent sulci, right Sylvian fissure, and compression of the ipsilateral temporal horn of the lateral ventricle. . Depending on the degree of differentiation of the components' tissues, teratoma is classified as mature or immature; mature teratoma can be identified based on the presence of fully differentiated elements from all three germinal layers.CNS GCTs  have an overall male predominance and in a series of 254 teratomas by Tapper and Lack, most of the patients were aged 21 years or less.As the tumor is often midline in the pineal region, obstructive hydrocephalus is the most common manifestation of CNS GCTs, and patients present with features of raised ICP. However, temporal/frontal lobe tumors are more frequently associated with seizures than other manifestations.MRI is considered the radiologic study of choice in the diagnosis and staging of the brain tumors, and the lesions of GCTs are hypointense on T1‐weighted hyperintense on T2‐weighted images, and non‐enhancing in contrast unless it has a cystic component. The presence of fatty tissue or multilocularity and calcifications is a characteristic predominant feature of teratoma.Histopathological examination of mature teratoma shows a well‐differentiated tissue with low mitotic activity, while immature teratoma shows hypercellular embryonic mesenchyme or primitive neuroectodermal elements mimicking fetal tissue that can be mixed with mature tissue elements with a rare incidence of malignant transformation.The treatment strategy of teratoma is still controversial. Gross total excision should be done whenever possible and the surgical approach depends on the location of the teratoma. Total excision of the tumor is the treatment of choice for mature teratomas as they are benign and usually radioresistant.The 10‐year survival is as high as 93% in one large series, which signifies an excellent prognosis of intracranial mature teratomas.4Intracranial germ cell tumors are rare with the temporal lobe being an unusual location. Moreover, mature teratoma presenting as a focal seizure is not common. Nonetheless, mature cystic teratoma should be considered as a differential diagnosis of intracranial tumors presenting with seizures in young patients.None to declare.GS and OS: did study concept, data collection, and surgical therapy for the patient. YR and SS: did writing – original draft preparation. BMS, SLB, and SB: did editing and writing. GS and OS: are senior authors and manuscript reviewers. All the authors read and approved the manuscript.Written informed consent was obtained from the patient's father 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.


Inflammatory cloacogenic polyps are a rare kind of benign polyp that is located in the anal transitional zone and rectum. We report the case of a 53-year-old male who underwent a diagnostic colonoscopy for a positive fecal immunochemical test. Two 7 mm polyps were found in the rectum with a pathological diagnosis of inflammatory cloacogenic polyp. The polyp was endoscopically resected. These polyps are associated with chronic inflammatory conditions such as Crohn’s disease and colorectal tumors. Because of malignant transformation potential, inflammatory cloacogenic polyps are endoscopically removed. Inflammatory cloacogenic polyps are a rare type of anorectal polyp that usually arise in the anal transitional zone . They shHistoryA 53-year-old female with a history of low-grade papillary tumor of the bladder, cigarette smoking, and alcohol abuse was seen in the gastroenterology clinic for colorectal cancer screening. She denied any current symptoms of abdominal pain, nausea, vomiting, hematemesis, melena, hematochezia, constipation, diarrhea, tenesmus, fecal urgency, fevers, chills, loss of appetite, or weight loss. She had no family history of gastrointestinal malignancy or inflammatory bowel disease.Physical examinationOn physical exam, we did not elicit any abdominal tenderness. There was no organomegaly or abdominal distention. Normal bowel sounds were heard all over the abdomen. A direct rectal exam was positive for dark-colored stool on the gloved finger.InvestigationsLaboratory studies revealed a hemoglobin 12.2 g/dl, hematocrit 40%, MCV 88.3 fL, platelet 368 k/uL. She tested positive on a FIT and subsequently underwent a diagnostic colonoscopy two weeks later. The colonoscopy revealed two sessile 7 mm polyps in the rectum (Figure Treatment planAfter polyp resection, the patient continues to be asymptomatic. Since cloacogenic polyps are benign, we did not pursue any further management. However, due to the presence of a tubular adenoma, the patient is scheduled to follow up in the gastroenterology clinic for endoscopic surveillance.Inflammatory cloacogenic polyps were first described by Lobert et al. in 1981 . An inflCloacogenic polyps are relatively more common in women between 40 and 60 years of age .The pathophysiology of cloacogenic polyps remains unclear. However, since it shares histological features with other causes of mucosal prolapse syndromes, such as solitary rectal ulcer syndrome, repeated mucosal ischemia and regeneration are postulated to play a role in pathogenesis ,5. SolitInflammatory cloacogenic polyps have been reported in adults with various gastrointestinal disorders, including Crohn’s disease, malabsorptive states, diverticulosis, colorectal tumors, and hemorrhoids. However, a causal relationship between the diseases and cloacogenic polyps has not been identified. Inflammatory cloacogenic polyps have also been associated with dysplastic lesions in the anus and anal intraepithelial neoplasia ,7. HumanEndoscopic or surgical resection of lesions in addition to a high fiber diet is the preferred treatment ,4. RoutiIn conclusion, inflammatory cloacogenic polyps are rare, benign, polypoidal lesions that can be found on colonoscopy. Since they can be mistaken for anorectal neoplasm or hemorrhoids, endoscopists need to be familiar with inflammatory cloacogenic polyps. Active surveillance after resection is necessary because of their high risk of recurrence and potential for malignant transformation.


Abdominal actinomycosis is a rare disease caused by a Gram-positive bacillus (Actinomyces). Liver manifestation is rare and, in patients with a history of cancer, differential diagnosis with secondary malignant disease can be difficult. Microbiological result is necessary for a correct diagnosis, though not always possible in preoperative workout. The authors present a case of hepatic actinomycosis that mimicked oncological disease and led to a more aggressive surgical approach. Actinomyces israelii is the most prevalent [Actinomycosis is a rare chronic granulomatous infection caused by a type of Gram-positive bacillus, Actinomyces. There are 13 different species described, six of them are pathologic [revalent . Cervicorevalent .Risk factors for hepatic actinomycosis include dental abscesses or caries, previous surgeries, alcohol abuse, diabetes, and hepatic lithiasis. This disease is more frequent in males in their fifth decade of life . We descA 52-year-old male with a history of high blood pressure, smoking, and moderate alcohol habits, was hospitalized for asymptomatic obstructive jaundice. Endoscopic retrograde cholangiopancreatography showed an ampullary tumor and biopsies revealed a high-grade dysplasia adenoma. After a multidisciplinary team decision, the patient underwent pyloric preservation pancreaticoduodenectomy. No events were registered and the patient was discharged on 10th postoperative day.Tumor was classified as poorly differentiated adenocarcinoma of the biliopancreatic confluence with lymph node metastasis - pT1G2N1 . Chemo Four years after the index surgery, patient was admitted to the hospital for cholangitis. Imaging study showed dilation of intrahepatic bile ducts, mostly on the left side, without a visible mass Figures , 1B. The9/L with 67.1% of neutrophils, hemoglobin was 13.6 g/dL, oxaloacetic transaminase was 32 U/L, alkaline phosphatase was 339 U/L, international normalized ratio (INR) was 1.2, and total bilirubin was 0.65 mg/dL.One year later, during follow-up, an increase of tumor markers (carbohydrate antigen {CA 19-9} of 1142 U/mL and carcinoembryonic antigen {CEA} of 6.46 ng/mL) was detected. Alpha-fetoprotein was not measured. The remaining analytical study was normal, leukocytes of 5.4x10Clinically, the patient was asymptomatic, without constitutional symptoms, anicteric, and with a normal physical examination. Abdominal CT was normal Figure . AbdominMultidisciplinary oncologic team decided for a surgical approach. The patient was submitted to an uneventful left hepatectomy. The patient was discharged on day nine. Histologic analysis revealed hepatic abscess compatible with actinomycosis. The patient was treated with doxycycline for six weeks. After the treatment tumor markers normalized and the patient was asymptomatic. The patient was kept under surveillance with CT and tumor markers every six months for two years.Isolated hepatic actinomycosis is a rare condition. Fever, weight loss, abdominal pain, and hepatic cystic lesion may suggest this entity as a differential diagnosis, although unspecific symptoms usually lead to late diagnosis . MicrobiIn the current case, etiological doubt was based on the past oncological history, namely, a metastasis from the carcinoma excised years ago. In fact, it is described that abdominal actinomycosis can resemble a malignant disease presenting with abdominal pain, asthenia, and weight loss . Less thIn up to 66% of cases, hepatic actinomycosis presents as a single hypodense lesion, as in this patient. More than 40% of imaging examinations may not distinguish from a malignant lesion; however, hepatic MRI typically shows an image characterized by hypointensity in T1 and hyperintensity in T2, as happened in this case. There are no specific findings in CT scan and ultrasonography, and positive blood cultures are extremely difficult to obtain. Tumor marker CA 19-9 might be increased, though levels are usually lower than seen in malignant disease [In this case, we were treating a young patient with a single, resectable liver lesion, with no evidence of disseminated disease that appears five years after resection of an ampullary tumor. The hypothesis of liver metastasis seemed less likely considering the elapsed time since the first surgery. Metastases from a digestive primary neoplasm were unlikely with a normal endoscopic study. The hypothesis of other primary liver tumors such as hepatocellular carcinoma was unlikely given the lesions’ imaging characteristics and the absence of liver cirrhosis . The finAt this moment, there are no surgical treatment guidelines for hepatic actinomycosis . TreatmeThis differential diagnosis cannot be neglected since, with the exception of the microbiological examination, there is no proper diagnostic tool for actinomycosis . There aIn this case, medical history was highly suggestive of a secondary lesion, so no preoperative biopsy was attempted, even though several infectious occurrences one year before should have raised the hypothesis of an infectious pathology in the differential diagnosis. Lack of symptomatology, high levels of tumor markers, doubtful MRI, and a positive PET scan deviated the suspicion towards malignancy leading to a more invasive approach.Hepatic actinomycosis is a rare entity characterized by indolent evolution and unspecific clinical and imaging features. Differential diagnoses include other liver infectious conditions, as well as malignant tumors . High level of suspicion is needed to adequate the best treatment option.


Gastrointestinal manifestations of leukemias have been well recognized. Typically, acute leukemias cause typhlitis or appendicitis more commonly than chronic leukemias. Our case points to appendicitis as possible manifestation of chronic myelogenous leukemia. Gastrointestinal manifestations of leukemias have been well recognized. Typically, acute leukemias cause typhlitis or appendicitis more commonly than chronic leukemias. Our case points to appendicitis as possible manifestation of chronic myelogenous leukemia. Chronic myelogenous leukemia (CML) is one of the myeloproliferative neoplasms that is usually diagnosed accidentally on routine complete blood count (CBC) because it is asymptomatic in most cases. However, it can cause multiple signs and symptoms and affect many organs, including the gastrointestinal system. We present a case of a 36‐year‐old patient presented with appendicitis found to have CML during his workup for his abdominal pain.Hematologic malignancies (HM) represent a burden on the healthcare system worldwide, mainly due to their prevalence among the pediatric population, and have always been well recognized among other types of cancer by the effective use of genetic analyses to establish the diagnosis, classification, and prognosis.9/L (4‐10 × 109/L).Here, we present a case of a 36‐year‐old male who presented with right lower abdominal pain and was accidentally found to have a very high white blood cell (WBC) count during routine workup 317.3 × 1029/L (4‐10 × 109/L)  showed moderate splenomegaly and dilated appendix, measuring 1.8 cm in maximum diameter with periappendiceal fat stranding and minimal free fluid in the right iliac fossa. No appendicoliths are noted, suggestive of acute appendicitis. He went for laparoscopic appendectomy and was found to have acute gangrenous appendicitis intraoperatively. The appendix was sent for histopathology, which revealed severe acute appendicitis Figures .t.After surgery, further investigation for leukocytosis was done. Peripheral smear showed normochromic normocytic anemia with severe leukocytosis, shift to left, and basophilia, suggestive of a myeloproliferative disorder. Cytogenetic and molecular studies were positive for an e14a2 BCR‐ABL1 gene fusion by single‐step RT‐PCR. No evidence of mutations within the JAK2 or CALR genes. Interphase fluorescence in situ hybridization (FISH) showed BCR/ABL1 rearrangement in 97% of nuclei. Bone marrow biopsy showed hypercellular bone marrow (~90%‐100% cellularity) with remarkable granulocytic hyperplasia with only 2% blast. Karyotype result showed 46,XY,The patient was started on hydroxyurea 1 g three times daily and rasburicase. After confirming the diagnosis, the patient preferred to get treatment in his home country and traveled back to India.3t, resulting in the fusion of the Abelson murine leukemia (ABL1) gene on chromosome 9 with the breakpoint cluster region (BCR) gene on chromosome 22 (BCR‐ABL1), and this rearrangement is known as the Philadelphia chromosome.Leukemias constitute one overwhelming branch of the hematologic malignancies and are mainly divided into lymphoblastic/lymphocytic and myeloblastic/ myelogenous leukemias. CML is one of the MPNs and accounts for 15% of diagnosed cases of leukemia in adults.In conclusion, leukemias whether acute or chronic may manifest in a variety of symptoms and conditions, and patients should be addressed with caution and in a comprehensive approach.None declared.RA: manuscript writing and literature review. EA: case presentation writing. LO: literature review. OE: pathology slides. MA: radiology slides. YM: literature review. MY: mentorship, manuscript writing, and literature review.Ethical Approval was obtained by Medical Research Center (MRC) under ID MRC‐04‐20‐977 on October 27th, 2020. The article processing charges were funded by Qatar National Library (QNL).


Adenocarcinoma in a Meckel’s diverticulum is rare and difficult to diagnose preoperatively. We report the first case of a metachronous Krukenberg tumor from adenocarcinoma in a Meckel’s diverticulum. A 45-year-old woman was admitted for recurrent abdominal pain. Computed tomography scan showed a lesion with contrast enhancement, and a Meckel’s diverticulum-associated tumor was suspected. Double-ballon enteroscopy revealed intestinal stenosis and biopsy showed adenocarcinoma. Operative findings showed a Meckel’s diverticulum with tumor. Histopathological evaluation revealed well-differentiated adenocarcinoma, interrupted by ectopic gastric mucosa, diagnosed as adenocarcinoma in a Meckel’s diverticulum. Two years postoperatively, a multi-cystic mass with contrast enhancement was observed in the pelvis on imaging evaluation and oophorectomy performed. Histological examination of the resected ovary showed proliferation of atypical glandular ducts, consistent with metastatic adenocarcinoma. This case demonstrates that adenocarcinoma in a Meckel’s diverticulum may result in distant metastases and requires appropriate follow-up. Meckel’s diverticulum is the most common anomaly of the gastrointestinal tract which is a true diverticulum. The prevalence is estimated to be ~1.2–2.0% of the population . Meckel’A 45-year-old woman was admitted with recurrent abdominal pain. She had no significant past medical history. Physical examination showed mild tenderness in the lower abdomen. Laboratory findings were within normal limits, except for serum carbohydrate antigen 19-9 (58 U/mL). Computed tomography (CT) scan showed a continuous lesion to the ileum with contrast enhancement . RetrogrImmunohistochemistry studies for caudal-type homeobox transcription factor 2 (CDX2), a marker for intestinal-type gastric cancer, showed strong expression in the adenocarcinoma . HoweverTwo years after resection, the patient had no symptoms, but the serum carbohydrate antigen 19-9 became elevated (73 U/mL). CT scan showed a multi-cystic mass in the pelvis with some contrast enhancement . GadolinTo the best of our knowledge, this is the first report of a metachronous Krukenberg tumor from adenocarcinoma in a Meckel’s diverticulum. The present patient confirms two important clinical points: (i) adenocarcinoma in a Meckel’s diverticulum may result in distant metastases and requires appropriate follow-up and (ii) double-balloon enteroscopic biopsy and contrast study are useful to establish a preoperative diagnosis.et al. [Meckel’s diverticulum is the most frequent congenital anomaly of the gastrointestinal tract, which is a true diverticulum resulting from incomplete obliteration of the omphalomesenteric duct . The preet al.  reportedThe preoperative diagnosis of tumors in Meckel’s diverticula is difficult. Previous reports demonstrate that tumors in Meckel’s diverticula were most often diagnosed after resection . Double-In conclusion, we report a patient with a metachronous Krukenberg tumor from an adenocarcinoma in a Meckel’s diverticulum. Intensive follow-up is necessary because of the aggressive nature of the tumor in this patient. Double-ballon enteroscopy was useful to preoperatively establish the diagnosis.