EWSR1), generating gene fusions. Extraskeletal ES mainly occurs in soft tissues; the urogenital tract is rarely affected, and ureteral localization is an exceptional event with only 4 cases described in the literature. Here we report the first Italian case of primary ES of the ureter, a 24-year-old young man with lower back pain and a narrowed left ureteral lumen on CT scan. ES of the urogenital tract is an almost unique condition with a nonspecific clinical presentation and a challenging diagnosis for pathologists. We encourage awareness of these exceptional events in the differential diagnosis of ureteral lesions in young patients.Ewing sarcoma (ES) is the second most common malignant bone tumor in children and has also been described in adults with highly aggressive behavior. ES belongs to the small round blue cell tumor family and presents the distinctive translocation of FET-ETS family genes (85% with  Ewing’s sarcoma (ES) is a malignant neoplasm with highly aggressive behavior . It is tin situ hybridization (FISH)—producing fusion genes that involve one member of the FET family of genes and a member of the E26 transformation-specific (ETS) family (EWSR1–FLI1 in 85%–90% of cases), encoding aberrant transcription factors  –11. As fHere we report the fifth case of primary ureteral ES and the first in Italy as a unique manifestation of the disease.A 24-year-old male presented to our institution with a 6-month history of left lower back pain. Five years before, he underwent surgery for varicocele and had a history of prostatitis. No relevant family medical history was documented. Abdominal US and CT scans of the urological tract with contrast medium were performed with the finding of a narrowed left ureteral lumen and ipsilateral hydronephrosis Figure 1Frozen sections showed a proliferation of round to ovoid epithelioid cells, arranged in solid sheets, with nuclear atypia and eosinophilic cytoplasm  tumor sections. Vysis LSI EWSR1 (22q12) Dual Color Break Apart Rearrangement Probe , a mixture of two probes that map the 5’ side of the EWSR1 gene (labeled in SpectrumOrange) and the 3’ side of the EWSR1 gene (labeled in SpectrumGreen), has been used. Signals were evaluated in at least 100 tumor nuclei per specimen. EWSR1 translocation was detected in 80% of cells in the tumor area , and cyclophosphamide (600 mg/m2) (VDC) alternating with five cycles of ifosfamide (3 g/m2) and etoposide (150 mg/m2) (IE). Subsequently, the patient underwent radicalization surgery with a partial ureterectomy and surrounding soft tissue resection, without residual tumor. After 15 months of follow-up, the patient was alive with no sign of recurrence or metastatic disease on CT total body and with programmed maintenance chemotherapy (VC×3/IE×2).Post-surgical CT total body and PET scan did not show any distant metastases. After a definitive diagnosis and multidisciplinary tumor board, systemic multiagent adjuvant chemotherapy was administered with four cycles of vincristine (2 mg), doxorubicin   . It is af cases) .ES usually affects the bone of children and young adults, but it can also arise in older patients and localize in extraskeletal sites, mainly in soft tissues . A revieEWSR1 and exon 6 of FLI, in keeping with the literature. Indeed, the most common breakpoints are exons 7 and 6 of EWSR1 and exons 7 and 5 of FLI1 (EWSR1–FLI1 fusion was identified only in one other patient by RT-PCR ( of FLI1 . Interesy RT-PCR . Only ony RT-PCR .In a clinical scenario with a young patient presenting with non-specific symptoms and a ureteral mass, in the absence of other primitive tumors, like in our case, the differential diagnosis should comprise a localization of either a germ cell tumor or a lymphoma. Blood markers and imaging to detect abdominal lymphadenopathy might be helpful in this setting. For our patient, the CT scan excluded further abdominal lesions, while blood markers were not tested.Although less frequent, a retroperitoneal soft tissue tumor should also be ruled out.In the suspected case of primary urothelial carcinoma of the ureter, our patient underwent surgery without a previous diagnosis. This is because the lesion could not be biopsied during diagnostic ureteroscopy due to lumen fibrotic obstruction. Therefore, an intraoperative evaluation was performed on the partial ureterectomy specimen. Frozen sections were somehow misleading since they suggested a urothelial neoplasm. Indeed, the tumor showed a more epithelioid morphology with round to ovoid cells with eosinophilic cytoplasm, growing in solid sheets with a fair degree of atypia. The urothelial nature was also consistent with site and clinical presentation, although in a young patient. The results of the intraoperative evaluation prompted the urologist to perform an uretero-ureteral anastomosis without radicalization.The clinical management after definitive diagnosis was challenging and required several multidisciplinary tumor boards to decide for a completion surgery and adjuvant chemotherapy. In daily practice, a preoperative diagnosis of ES is of paramount importance to the oncologist since these tumors—of any site—undergo standardized protocols with preoperative neoadjuvant chemotherapy . Indeed,Remarkably, due to the lack of a preoperative histological diagnosis, three of the four previously reported cases received chemotherapy only after surgery –14. In oTo the best of our knowledge, this is the fifth reported case of primary ureteral ES and the first in Italy. ES of the urogenital tract is an exceptional condition with nonspecific clinical presentation and a challenging diagnosis, with a possible impact on oncological management due to the common lack of preoperative histological information precluding neoadjuvant treatment protocols. We encourage awareness of these exceptional events in the differential diagnosis of ureteral lesions in young patients.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the patient for the publication of any potentially identifiable images or data included in this article.MV and PC conceived, designed, wrote, and revised the final manuscript. LD reviewed the literature, provided figures and tables, and revised the final manuscript. MC and GE reviewed the histological slides, critically revised the work, and approved the final manuscript. NR performed molecular analyses. AS performed surgery. AB followed the patient and provided clinical data. All authors contributed to the article and approved the submitted version.


Neurofibromas are benign tumors. They are known to be a manifestation of von Recklinghausen’s disease (neurofibromatosis type 1) and tend to be generalized and rarely solitary. In this report, we present a case of solitary neurofibroma in the maxillary gingiva. A 39-year-old woman presented with a chief complaint of swollen gingiva. There were no special findings in her medical or family history. After a biopsy was performed under local anesthesia and a diagnosis of neurofibroma was made, tumor resection was performed under general anesthesia. The patient’s progress was good, with no recurrence. Neurofibromas are benign tumors. The 2017 World Health Organization classification defines it as a benign peripheral nerve sheath tumor composed of a mixture of palisade cells, neuropericytes, fibroblasts and axons . In thisA 39-year-old woman presented with a chief complaint of swollen left maxillary gingiva. She had been aware of the swollen gingiva for 6 years but had neglected it because it was not painful. Her family dentist recommended a thorough examination, and she visited our department for her first visit. There were no special findings in her medical or family history. The extraoral examination was normal, but intraoral examination revealed a bulge in the buccal gingiva of the left maxillary canine . The mucThere was no evidence of bony swelling or resorption on computed tomography (data not shown). On magnetic resonance imaging, contrast-enhanced T1-weighted images showed a mass with low signal , measuriThe specimens were collected from the gingival cheek transition area. The gingival mucosa was normal and contained a yellow mass that was full, elastic and hard. The specimen was a yellowish-white, substantial, inelastic mass. At the time of biopsy, the histopathological diagnosis was neurofibroma.Since there was no other sign of neurofibromatosis, a solitary neurofibroma was diagnosed. The patient underwent tumor resection under general anesthesia. The resection included the subperiosteum, with a surgical margin of 5 mm. The surrounding bone was smooth, stained with pyoctanine blue solution and one layer was removed. An artificial dermis was applied to the raw surface. The explant was pale yellow and covered with a membrane . The patThe lesion was a poorly circumscribed tumor proliferating in the submucosal area . The tumNeurofibromatosis type 1 is a hereditary neurocutaneous disease with significant effects on the nervous system, eyes, skin and bones . The cliNo racial differences have been reported for neurofibromatosis type 1, but 14 of 16 cases we collected were reported from Asian countries, suggesting a regional or racial predilection in solitary neurofibroma. In addition, the diagnosis of solitary neurofibroma requires a clinical rejection of neurofibromatosis type 1. However, it is difficult to rule out neurofibromatosis type 1, especially in young people because some of neurofibromatosis type 1 are known to occur without obvious genetic mutations or familial effects, and the main symptoms may occur at different times , 5. This


Mucopolysaccharidosis VI is a genetic disorder affecting multiple organs with sundry clinical presentations. The main etiological factor reflects the disturbances in mucopolysaccharide metabolism leading to deposition of acid mucopolysaccharide in various tissues. The pathognomonic features of the disease include a large head, short neck, corneal opacity, open mouth associated with an enlarged tongue, enlargement of the skull, and long anteroposterior dimension with unerupted dentition, dentigerous cyst-like follicles, condylar defects, and gingival hyperplasia. An 18-year-old boy with Maroteaux-Lamy syndrome (mucopolysaccharidosis type VI) is described in this article, emphasizing the oral manifestations and radiographic illustration of lesions in the jaws. It also emphasizes the essential role of cone-beam computed tomography to identify and analyze multicentric pathologies in the jaws. Mucopolysaccharidosis: MPS VI (Maroteaux-Lamy syndrome) is a rare genetic disorder caused by glycosaminoglycans (GAG) storage enzyme deficiency: N-acetylgalactosamine sulfatase. Normally this enzyme is found in the skin, tendons, blood vessels, airways, and heart valves , 2. The ARSB mutation culminates in excessive accumulation of GAG and DS in the cells, which triggers the tumor necrosis factor (TNF) pathway . This elElevated GAGs in the urine and high DS concentrations are markers of disease activity but alone, these are not diagnostic methods. The criterion for the diagnosis is based on the estimation of ARSB enzyme activity confirmed in cultured fibroblasts or isolated leukocytes . It is estimated that the urinary score of GAG is directly proportional to the progression of the disease . Though various treatments have been mentioned in the literature, an interdisciplinary approach is a prerequisite for the management of patients with MPS VI , 9.The objective of this article was to report the case of an 18-year-old male patient in Western Uttar Pradesh, India, seeking endodontic treatment for painful swelling of maxillary anterior teeth. He was diagnosed with MPS VI, focusing on the general characteristics of MPS VI and radiological findings. It also emphasizes the importance of radiographic examination of all unerupted teeth, using Cone Beam Computed Tomography (CBCT) to better delineate the extent of the lesion and its relationship to adjacent anatomical structures.An 18-year-old male patient reported to the Department of Oral Medicine and Radiology, IDST Modinagar, seeking treatment for orofacial swelling and purulent discharge in left upper front teeth for 6 months. The personal history revealed that both his parents were middle-aged in the fourth decade of life. His mental status was normal; however, cardiac and broncho-pulmonary insufficiency and mild hepato-splenomegaly were noted. The patient had a large head with corneal clouding in the left eye, flattened nasal bridge, bushy eyebrows, and hypertelorism . IntraorThe patient had a radiological examination of the local lesion concerning 23, and intraoral periapical (IOPA) revealed two radiolucencies. A smaller, well-defined radiolucency at the alveolar crest measuring approximately 3 mm × 2.5 mm in greatest diameter between the left lateral incisor and first premolar. Larger radiolucency was associated with a partial image of radiopacity, causing deflection of roots of the left lateral incisor and first premolar suggestive of cystic lesion associated with an unerupted impacted tooth . TherefoCBCT was acquired to assess the details of lesions manifested in both jaws. The tomographic images were obtained with a CS 9300 scanner at 0.18 mm × 0.18 mm × 0.18 mm resolution  using 90 kV, 15 mA, and 12 seconds of exposure time. The exposure settings were automatically optimized for the patient's size. The voxel size was 300 micrometers. The images were evaluated by an experienced oral and maxillofacial radiologist using CS 3D imaging software  in a dimly lit room on a 17 inch Dell monitor (Intel R Xenon R). CBCT images showed multiple well-defined expansile lesions described below , 6 and 7Second lesion measuring 5.5 cm × 3.6 cm × 4.3 cm in AP-T-SI dimensions, respectively, in the left maxilla, including the left hard palate and the left maxillary sinus extending anteriorly across the midline to the 12 region. The lesion involved the periapical region of the teeth 12 until 28 and occupied the entire left maxillary sinus cavity. Tooth 23 was seen within the lesion in the anterior superior third of the left maxillary sinus. The margins were well defined, thin corticated, and scalloped. Partial septations were also noted in the inferior third of the lesion. The internal structure was completely radiolucent with no internal calcifications. Apical root resorption was seen in teeth 26, 25, 11, and 12. The lesion involved the nasopalatine canal with effacement of the cortical outlines. Thinning and mild expansion of the labio-buccal cortex was present. In addition, thinning and expansion of the adjacent palatal cortex were noted.Third lesion measuring 3.14 cm × 1.2 cm × 3.4 cm in AP-T-SI dimensions, respectively, in the right posterior mandible and right mandibular ramus extending from the 47 region till the posterior third of the right mandibular ramus. Supero-inferiorly the lesion involved the entire occluso-apical extent of the alveolus in the 48 region. It involved the buccal peri-radicular region of tooth 47. Thin corticated scalloped margins were seen. Internal contents were completely radiolucent with no internal calcifications/septations. The developing tooth 48 was displaced inferiorly, and the lesion appeared attached to the CEJ of the tooth. Effacement was seen on the crest of the alveolus in the 48 region. There was a thinning and mild expansion of the adjacent buccal and lingual cortices. There was a medial and inferior displacement of the adjacent right inferior alveolar canal. The vertical segment of the canal was displaced posteriorly.Fourth lesion measuring 1.2 cm × 3.1 cm × 3.0 cm in AP-T-SI dimensions respectively, in the anterior and left parasymphysis region, extending from the 34 region across the midline till the 43 region. The lesion involved the pericoronal region of the mesioangularly impacted tooth 33. The lesion also involved the apical alveolar-basal bone and the periapical region of teeth 32, 34. It extended into the interdental alveolar bone between teeth 32–34 with effacement of the alveolus crest. Thin corticated scalloped margins were observed. Internal contents were completely radiolucent with no internal calcifications/septations. There was a thinning and mild expansion of the adjacent labio-buccal cortex. Mild thinning of the lingual cortex and inferior cortex was observed. Apical root resorption was seen in teeth 34, 32. Fifth lesion measuring 1.5 cm × 1.1 cm × 1.4 cm in AP-T-SI dimensions respectively, involving the interdental alveolar bone between teeth 36–37. The lesion involved the distal-lingual peri-radicular region of the distal root of 36 and the mesial-lingual peri-radicular region of the mesial root 37. The margins were thin corticated with completely radiolucent internal contents. There was a thinning and mild expansion of the adjacent lingual cortex. Effacement was seen along the crest of the alveolus lingual to the roots of teeth 36–37. The lesion was caudal to the left inferior alveolar canal.Sixth lesion measuring 3.5 cm × 1.1 cm × 4.6 cm in AP-T-SI dimensions respectively, in the left posterior mandible and left mandibular ramus extending from the 37 region till the posterior border of the left mandibular ramus. Supero-inferiorly the lesion involved the entire occluso-apical extent of the alveolus-basal bone and the craniocaudal extent of the ramus. It involved the buccal and distal peri-radicular region of tooth 37. The developing tooth 38 was displaced inferiorly, and the lesion appeared attached to the CEJ of the tooth. Thin corticated scalloped margins were observed. Internal contents were completely radiolucent with no internal calcifications/septations. Effacement was seen on the crest of the alveolus in the 38 region. There was thinning of the adjacent buccal and lingual  cortices and anterior cortex of the ramus. A mild expansion was seen of the medial cortex of the ramus. There was a medial and inferior displacement of the adjacent left inferior alveolar canal. The vertical segment of the canal was displaced posteriorly.Clinical and radiographic findings indicated a syndrome such as cleidocranial dysplasia, basal cell nevus syndrome, Klippel-Feil syndrome, Maroteaux-Lamy syndrome suffering from multiple dentigerous cysts. The patient was advised to take a posteroanterior chest x-ray and anteroposterior skull and neck radiograph to detect osseous abnormalities in these syndromes, but the radiographs appeared normal. On laboratory investigations, thyroid profile, serum calcium, phosphorus levels, and 25-hydroxy cholecalciferol levels were normal. Metabolic screening tests for GAGs in the urine and aryl sulfate B assay were advised to determine Maroteaux-Lamy syndrome. GAG concentration in the urine was 134 mg GAG/g creatinine . Enzyme assay revealed a deficiency of aryl sulfate B that was 83.26nmol/mg proton/h . The deranged values indicated the presence of Maroteaux-Lamy syndrome. We explained to the patient the importance of identifying the variant in the family through genetic testing. Genetic testing was offered to both parents to avoid such complications in future pregnancies but could not be performed because of the poor affordability of parents. The patient was notified of the diagnosis and opted not to pursue treatment. No noticeable clinical changes were observed at a 6-month follow-up appointment.Mucopolysaccharidosis: MPS VI (Maroteaux-Lamy syndrome) is a rare syndrome usually having an autosomal recessive inheritance pattern . The maiThe presence of average intelligence, prominent metachromatic inclusions in leukocytes, and deficient activity of ARSB differentiate this syndrome from other mucopolysaccharidoses , 9. SystExtraoral findings include a large and prominent forehead, marked supraorbital ridges, temporal bulge, short neck, corneal opacity, enlarged skull, and a long anteroposterior dimension –11. IntrAnother case report described oral manifestations of Maroteaux Lammy syndrome in an 11-year-old boy with similar findings . Though Radiographic examinations are essential in the case of patients with unerupted teeth. However, a panoramic radiograph may be used in extensive lesions. CBCT is indicated for better delineation of the extent of the lesion and for establishing the lesion's relationship with adjacent anatomical structures supplemented with skull radiographs .et al. described these osteolytic lesions as two types: the cystic type, which is destructive in nature as observed in the present case. The other type that rarely causes displacement and is limited to the crown of the unerupted tooth is the connective tissue type, mainly containing hyaluronic acid, a glycosaminoglycan that may lead to this disorder , and the collected data was accessible to the authors only (IDST/IERBC/CR/002/20).The written and oral consent of the patient's mother was obtained before the documentation of the case.SR, DM contributed to conceptualizing. AM, AJ contributed to the methodology. AM, DG contributed to writing the original draft. AV, AH contributed to editing the manuscript. DM, AV contributed to data curation.


An 80-year-old man was referred to our hospital because of epigastric pain. Abdominal computed tomography revealed a well-defined circular intra-abdominal mass in the gastro-pancreatic region measuring 15 mm in diameter. After 6 months, the mass lesion was growing with mild enhancement, and weaker enhancement was found in the lower half of the mass on contrast-enhanced computed tomography. The mass lesion touched the stomach, whereas adipose tissue appeared to intervene between the mass and pancreas. On magnetic resonance imaging, the well-defined mass lesion had isointensity to muscle on T1-weighted imaging, slight hyperintensity to muscle on T2-weighted imaging, which indicated a rich fibrous tumor. Under general anesthesia, the patient underwent open surgery. Intraoperatively, the tumor was separated from the stomach and firmly attached to the pancreas. Therefore, we performed a distal pancreatomy with splenic resection. Pathological diagnosis was desmoid-type fibromatosis in the retroperitoneum, and the tumor margin was attached to the pancreas, splenic artery, and splenic vein. Since there are few reports of desmoid-type fibromatosis occurring in the retroperitoneum of the gastropancreatic region, it is difficult to distinguish from other soft tissue tumors and to identify the tumor origin. Close observation by radiological re-valuation was a useful option. Magnetic resonance imaging signals and an enhanced pattern may help distinguish a desmoid-type fibromatosis from other soft tissue tumors. A desmoid-type fibromatosis that is well-defined in radiological findings may infiltrate the surrounding organs with gross or pathological analyses. A desmoid-type fibromatosis (DTF) is a tumor-like proliferating fibrous tissue disorder that originates from the connective tissue of the fascia or aponeurosis Here, we present a rare case of a slow growing DTF arising from the retroperitoneum in the gastropancreatic region. Radiologically, the lesion did not infiltrate the surrounding organs. However, intraoperative findings revealed a distal pancreatomy that required resection, and the tumor margin was pathologically in contact with the pancreas, splenic artery, and splenic vein, which indicated a dissociation between the preoperative findings and postoperative findings. We report the clinical experience of this case for improvement of radiological diagnoses.An 80-year-old man was referred to our hospital because of epigastric pain. He had a medical history of hypertension and hyperlipidemia. His surgical history included endoscopic mucosal resection for early gastric cancer. Laboratory assessment was within normal limits including tumor markers. Abdominal computed tomography (CT) revealed a well-defined circular intra-abdominal mass in the gastropancreatic region measuring 15 mm in diameter . After 4We recommended surgical resection to the patient for the following reasons: (1) a definitive diagnosis was difficult radiologically, (2) the tumor grew gradually within 6 months, and (3) the tumor could be resected without organ resection.Under general anesthesia, the patient underwent open surgery. Intraoperatively, the tumor was separated from the stomach and firmly attached to the pancreas. Therefore, we performed a distal pancreatomy with splenic resection. The intraoperative frozen section diagnosis denied a pancreatic invasive ductal adenocarcinoma. On gross section, the tumor had an irregular shape and was 15 × 20 × 35 mm; the cut surface showed a mixture of white and dark red colors. Histological findings showed abnormal proliferation of spindle-shaped cells with a slightly thick fibrous structure without strong atypia . MicroscPostoperatively, the patient developed a pancreatic fistula and underwent CT-guided drainage 9 days after surgery. Under local anesthesia in a right lateral position, an 8.5Fr J type catheter was inserted. The pancreatic fistula was treated with drainage and medical therapy. The patient was discharged 48 days after surgery.A DTF is a locally aggressive fibroblastic proliferation that is characterized by a variable and sometimes unpredictable clinical course A DTF is a distinct rare entity that accounts for 0.03% of all tumors and 3% of all soft tissue tumors, with an incidence of 2-4 cases per million per year in the general population Imaging plays an important role in the management of a DTF; the most common imaging modalities are CT and MRI, as well as ultrasonography in select cases Regarding the radiological CT findings in our case, we suspected the mass lesion was a metastatic lymph node, malignant lymphoma, Castleman's disease, or gastrointestinal stromal tumor. The MRI findings indicated a rich fibrous tumor such as Castleman's disease, a solitary fibroma, and a DTF. Among them, the enhanced patterns were not similar to Castleman's disease and a solitary fibroma; however, we could not confirm a diagnosis of DTF. The diagnostic accuracy of “first impression” based on MRI was reported to be higher than that based on CT The most common origin of an intra-abdominal DTF is from mesenteric or retroperitoneum connective tissue According to the desmoid tumor working group, the treatment options for intra-abdominal or retroperitoneal DTFs are surgery, radiotherapy, and medical therapy The recurrence rate is 15%-30% for an intra-abdominal DTFThis is a single case report, which is a limitation of this study. There are only a few reports of DTF in the retroperitoneum of the gastropancreatic region, making it indistinguishable from other tumors. Thus, accumulation of cases is necessary to improve imaging diagnoses of DTFs in the retroperitoneum of the gastropancreatic region.The clinical course of DTFs is variable and unpredictable; therefore, a multidisciplinary approach personalized to the individual patient is required. Close observation by radiological re-valuation was a useful option for a well-defined tumor in the retroperitoneum of the gastropancreatic region, which was pathologically diagnosed as a DTF after surgical resection. MRI signals and an enhanced pattern may help distinguish a DTF from other soft tissue tumors. A DTF that is well-defined in radiological findings may infiltrate the surrounding organs with gross or pathological analyses.The written informed consent was obtained for publication.


To date, only 25 cases of cerebral infarction following a bee or wasp sting have been reported. Due to its rarity, undefined pathogenesis, and unique clinical features, we report a case of a 62-year-old man with progressive cerebral infarction following bee stings, possibly related to vasospasm. Furthermore, we review relevant literature on stroke following bee or wasp stings.A 62-year-old retired male presented with progressive ischemic stroke after bee stings to the ear and face. Initial magnetic resonance imaging of the brain showed small punctate infarcts in the left medulla oblongata. Head and neck computed tomography angiography showed significant stenosis in the basilar artery and occlusion in the left V4 vertebral artery. The patient received intravenous alteplase (0.9 mg/kg) without symptomatic improvement. Digital subtraction angiography later demonstrated additional near occlusion in the left posterior cerebral artery (PCA). Thrombectomy was considered initially but was aborted due to hemodynamic instability. Repeated CT brain after 24 h showed acute infarcts in the left parieto-occipital region and left thalamus. The near occluded PCA was found to be patent again on magnetic resonance angiography (MRA) 25 days later. This reversibility suggests that vasospasm may have been the underlying mechanism. Unfortunately, the patient had persistent significant neurological deficits after rehabilitation one year later.Cerebral infarction following bee stings is rare. There are several proposed pathophysiological mechanisms. While the natural course of this phenomenon is not well characterized, early diagnosis and treatment are essential. Furthermore, it is important to establish standardized care procedures for this unique entity. Typical allergic symptoms include mild urticaria to severe anaphylaxis. Various unusual reactions after bee stings have been reported, involving neurological, renal, cardiac, pulmonary, and ocular systems . To date2A 62-year-old retired male with only mild hypertension was brought to the emergency department 1 h after bee stings to the ear and face. The initial symptoms were nausea, vomiting, and headache. On examination, he was found to have dysarthria, bulbar weakness, and right hemiparesis involving the arm and leg. The National Institutes of Health Stroke Scale score was 5. His blood pressure on presentation was 120/77 mmHg. The electrocardiogram showed sinus rhythm with a heart rate of 72 beats per minute. His initial non-contrast head CT was negative for hemorrhage or any acute processes. To treat a possible allergic reaction, he was given intravenous methylprednisolone and calcium gluconate. Given the possibility of acute stroke, intravenous alteplase (0.9 mg/kg) was also administered. Unfortunately, his symptoms did not improve.Magnetic resonance imaging (MRI) of the brain was performed, which showed acute infarcts in the left medulla oblongata .Head and neck CT angiography (CTA) was performed, which showed significant stenosis in the basilar artery, and occlusion in the V4 segment of the left vertebral artery . DigitalHe had a normal hepatorenal function, interleukin-6, fibrinogen, urine analysis, erythrocyte sedimentation rate, and coagulation testing. He was discharged to an acute rehabilitation facility. Unfortunately, at a one-year clinical follow-up, he had persistent neurologic deficits. He required percutaneous endoscopic gastrostomy due to dysphasia and tracheostomy. He was also unable to ambulate independently.Ethical approval: The research related to human use has been complied with all the relevant national regulations, institutional policies and in accordance with the tenets of the Helsinki Declaration, and has been approved by the authors’ institutional review board or equivalent committee.Informed consent: Informed consent has been obtained from all individuals included in this study.3Adverse reactions to bee or wasp stings may present with neurological, cardiovascular, renal, pulmonary, and ocular symptoms ,2. StingMost patients had favorable outcomes with immediate treatment; however, some had poor outcomes and four patients died ,7,22,24.There are no specific guidelines or expert consensus on treating cerebral infarction associated with bee or wasp stings due to limited reported cases and variations in presentations. According to reported cases, it is reasonable to consider treatment with epinephrine, methylprednisolone, antihistamine, and other suitable anti-allergic drugs at an early stage. Bees generally leave their stinging apparatuses in patients’ lesions after envenomation; the Vespidae attached to the sting site often persistently inject venom. Prompt removal of such stinging apparatuses is likely beneficial . Two patTo date, the pathophysiological mechanisms of bee or wasp sting-associated cerebral infarction are not fully elucidated. Wasps are members of the order Hymenoptera, suborder apocrita. Sensitization to wasp venom requires only a few stings. Also, symptoms may occur after a single string , includi1) Immune system hyper-functionality: Riggs et al.  reported2) Global cerebral hypoperfusion: Hypotension post-sting may be attributed to histamine and prostaglandin-2 induction . Vidhate3) Retrograde stimulation of the superior cervical ganglion: ,5 Riggs 4) Disseminated intravascular coagulation (DIC): Jain  proposed5) Vasoconstriction: Bee venom contains vasoactive peptides such as thromboxane, leukotrienes, and other vasoactive mediators, causing vasoconstriction that can lead to ischemic stroke ,21. The Postulated mechanisms include the following:4In summary, cerebral infarction following bee stings is rare. Various mechanisms have been proposed but there is no consensus on the most common pathophysiology or management approach. Further experiments, including in animal models, would be of great interest to elucidate the cellular responses to the venom that lead to vascular compromise and cerebral ischemia.


Pathological examination ruled out parathyromatosis and lung malignancy; despite its location outside the anticipated embryonic pathway, pathology revealed the presence of an ectopic parathyroid adenoma. After the surgery, serum parathyroid hormone and calcium levels decreased, and hypoparathyroidism was corrected with calcium carbonate and calcitriol.Primary hyperparathyroidism (PHPT) is the most prevalent cause of hypercalcemia, affecting 0.3% of the population. The only curative procedure is parathyroidectomy. Persistent PHPT occurs in 4.7 percent of patients, even in the most skilled hands. Ectopic adenomas are challenging to localize before and during surgery and frequently result in persistent PHPT. We presented a case with persistent PHPT due to lung parathyroid adenoma that was successfully resected with video-assisted thoracoscopic surgery. A 55-year-old female patient was admitted to our endocrinology clinic with persistent PHPT after four neck explorations over 16 years. The last  Primary hyperparathyroidism is defined by increased parathyroid hormone secretion resulting from parathyroid adenoma, hyperplasia, or, in rare cases, parathyroid malignancy . ParathyA 55-year-old woman with a history of chronic generalized pain and recurrent kidney stones was referred to our center in April 2021. In the last 16 years, the patient has undergone neck (exploration) surgery four times, however, PHPT has not been cured Figure 1She underwent orthopedic surgery sixteen years ago due to a pathological right tibia fracture that resulted in a nonunion. The patient had elevated levels of intact parathyroid hormone (iPTH) and serum calcium following surgery, despite the lack of additional diagnostic tests Figure 2The patient had been symptom-free for seven years prior to presenting with generalized body pain. She underwent a second neck exploration due to elevated serum iPTH and calcium levels and increased uptake in the left upper side of the neck on the 99mTc Sestamibi (MIBI) scan Figure 1She had a fracture in her left wrist seven years later with a poor healing process; the serum calcium level was up to 10.2 mg/dL and iPTH 106 pg/mL, 24-hour urinary calcium level was 214 mg. A SPECT-CT scan showed neither an ectopic mediastinal parathyroid adenoma nor any neck lesion. She underwent additional neck surgery due to high serum iPTH levels, while serum calcium levels remained in high normal reference ranges or slightly higher on two occasions. In the prior surgery report, the surgeon claimed that all four parathyroid glands were removed; however, pathology indicated just one hyperplastic parathyroid gland, and all excised tissue was reported to be nonspecific. Intact PTH levels remained high in all subsequent postoperative visits Figure 2Her laboratory exam in March 2021 showed a serum calcium level of 10.2 mg/dL, high serum iPTH, and normal Vitamin D level. The SPECT-CT scan showed nothing suggestive of parathyroid adenoma, neither in the neck nor in the mediastinum, but a solitary MIBI avid nodule was reported in the lower lobe of the right lung (11 x 5 mm) and was proposed to be ruled out for malignancy Figure 4PHPT is associated with a number of long-term consequences, including decreased bone mineral density and an increased risk of fracture nephrolithiasis, kidney failure, and neuropsychiatric disorders such as weakness, easy fatiguability, drowsiness, and depression . PHPT isParathyromatosis is an uncommon but challenging cause of persistent PHPT; multiple nodules of benign hyperfunctioning parathyroid tissue are characterized as being spread throughout the neck and mediastinum. There are two possible explanations for the condition. The first hypothesis postulates that parathyromatosis occurs due to parathyroid tissue spillage and seeding inside the operative field during parathyroid surgery and accounts for most cases reported in the literature. The second hypothesis is that physiological stimuli induce hyperplasia of preexisting parathyroid rests of embryological origin. The diagnosis of parathyromatosis did not apply to our patient due to the location of the adenoma in the lungs and the pathological characteristics. Another factor contributing to parathyroid surgery failure is the presence of PHPT in multiple endocrine neoplasia (MEN) syndromes. PHPT associated with MEN syndrome is an asymmetric multiglandular, multifocal disorder; most cases are benign tumors represented by parathyroid hyperplasia or adenoma ; it occuParathyroid adenoma is typically found in the neck region, particularly on the thyroid’s posterior capsule or in ectopic places . A prevaDiagnostic imaging should be conducted to localize the source of iPTH secretion. First-line localization procedures are neck ultrasonography, magnetic resonance imaging, computed tomography, and MIBI scan with or without SPECT/CT. The PET/CT is another imaging modality usually utilized in patients with persistent PHPT who are difficult to -localize . Two or There are two limitations in the presented case study. First, not using 11C-Choline or 18F-fluorocholine PET imaging, as recommended for examination of parathyroid tissue, due to lack of access. Second, there was no genetic examination of the hyperparathyroidism jaw tumor syndrome, which is one of the syndromes associated with primary hyperparathyroidism, even though the presented patient did not have other clinical manifestations of the syndrome, such as multiple ossifying fibromas of the maxilla and mandible, as well as renal and uterine tumors.In conclusion, to our knowledge, this is the first case of persistent PHPT with parathyroid adenoma in the lung that has been documented in the literature. VATS was conducted safely and without complications, and blood calcium and iPTH levels immediately improved following the procedure. Aside from the conventional locations from the neck to the mediastinum, atypical regions should be assessed for persistent PHPT to avoid long-term complications and additional procedures.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by Research Institute for Endocrine Sciences of Shahid Beheshti University of Medical Sciences. The patient provided their written informed consent to participate in this study.MV, AE, AA, and AZ were responsible for data collection and manuscript preparation. All authors approved this manuscript prior to submission. All authors contributed to the article and approved the submitted version.


A 11-month-old male intact Shar-Pei (26. 5 kg) was presented for a bilateral sublingual swelling of 4 months duration. The exploration of the oral cavity highlighted the presence of bilateral sublingual swellings, primarily consistent with bilateral ranula. The bilateral disease was treated with two subsequent surgeries 4 weeks apart. During the surgery, after removing an elliptical portion of the mucosa of the sublingual swelling, the presence of gelatinous tissue was visualized, and no saliva was present. The result of histological exam was oral mucinosis. At the subsequent follow-up the dog was in excellent conditions, without any symptoms. 1 month after the last operation, the dog underwent a visit in sedation to better evaluate the oral cavity. Both surgical sites were well-healed and without the presence of relapses. Upon 8 months follow-up the patient remained free of disease. This is the first reported case of oral mucinosis in sublingual mucosa in dogs. In this case the surgical treatment was curative. Oral focal mucinosis (OFM) is a rare clinic-pathological condition histologically similar to focal skin mucinosis and thus, OFM is considered as the oral counterpart of cutaneous focal mucinosis , 2.Cutaneous mucinosis refers to an excessive deposition of mucinous substance in the dermis that clinically manifests as a thickening of the skin or as a vesicular appearance , 4.A generalized cutaneous mucinosis, of likely genetic origin, occurs primarily in Shar-pei dogs, giving them its characteristic appearance . In otheIn human, OFM was first described and named by Tomich in 1974 .Clinically, oral lesions often present as a small (< 1 cm) submucosal, single, pink, and slow-growing asymptomatic nodule containing gelatinous material , 9. The OFM has already been reported in literature in a dog as a nodular lesion on the buccal mucosa . This isWritten informed consent was obtained from the owner for the publication of this case report.A 11-month-old male intact Shar-Pei (26.5 kg) was presented for a bilateral sublingual swelling of 4 months duration. The owners noticed that the was unable to masticate an pick up the food.The patient had undergone bilateral entropion reduction surgery 3 months earlier. The dog was otherwise healthy with no known systemic diseases, and he was up to date on vaccines and flea and tick prevention. Two months prior to presentation, the dog was prescribed an anti-edema drug (Seaprose s) by the referring veterinary, without improvement.At presentation, physical examination and vital parameters were within normal limits.The exploration of the oral cavity with the patient under sedation highlighted the presence of bilateral sublingual swellings of soft and fluctuating consistency, primarily consistent with bilateral ranula.Preoperative hematology and blood chemistry test were within normal limits. An ultrasound examination of the neck region was also performed to assess the integrity of the salivary glands. Chest x-rays were also performed. No remarkable alterations were observed.During the surgery, with the patient in left lateral recumbency , after wThe removed mucosa and the gelatinous tissue were submitted for histological examination.A post-operative therapy including antibiotic , NSAID  and anti-acid  was administered.Histologically, the subepithelial tissue was thickened and collagen bundles were distorted and widely separated by abundant, wispy, amphophilic material (mucin) and increased clear space (edema), admixed with granulation tissue, few lymphocytes and plasma cells. The overlying mucosal epithelium was mildly hyperplastic. The lacy material interpreted as mucin stained pale blue with alcian blue, whereas Periodic-acid Schiff reaction was negative, confirming its acid mucopolysaccharide composition .Seven days later, at the follow-up visit, the dog showed no symptoms. The animal no longer exhibited difficulties in grasping and chewing food.One month after the first operation, the dog underwent surgical removal of the contralateral swelling with the same procedure . The sitIn the light of the histological diagnosis, two skin biopsies with 4 mm Baker's punch were also performed.The histological examination of the oral lesion confirmed the previous diagnosis. The examination of skin biopsies revealed a mild dermal thickening due to increased accumulation of pale-staining, fibrillar to granular mucin strands, thereby allowing to diagnose dermal mucinosis as well. At the subsequent follow-up the dog was in excellent condition, without any symptoms.Under sedation oral examination performed 1 month after the last surgery revealed no evidence of relapse and surgical sites looked great. The patient remained disease free upon 8 months follow-up.To our knowledge, only one case of OFM in dogs has been reported in literature. However, the lesion was a solitary asymptomatic nodule on the buccal mucosa . The dogIn human medicine the term OFM was first introduced in 1974 by Tomich, et al. .Studies have suggested that it is caused by the overproduction of HA by fibroblasts , 13. AltIn human the most common intraoral sites of OFM are gingiva (58.2%), palate (15.3%), and alveolar ridge mucosa (8.2%), while in our case the lesion is found in the sublingual mucosa .OFM manifests as a localized, sessile or pedunculated mucosal overgrowth, varying from few millimeters to 2 cm in size. They clinically mimic pyogenic granuloma, peripheral giant cell granuloma, peripheral ossifying fibroma, traumatic fibroma or focal gingival hyperplasia . We can In our case, the lesion occurred as a bilateral sublingual swelling, similar in appearance to a ranula. While in the ranula the content is made up of saliva, in the current case there was the presence of gelatinous tissue, which was histologically and histochemically consistent with mucoprotein material rich in acid mucopolysaccharides. Before surgery it might have been advisable to carry out an FNA but the patient's age, anamnesis, localization, clinical symptoms and the findings of the oral visit convinced us to think of the presence of a bilateral ranula.The surgery of sublingual mucocele entails the removal of the affected salivary gland in addition to marsupialitation. In this case, as ranula was the diagnosis at the time of the first surgery, we would have had to remove the salivary gland before marsupialization. But, after the ultrasound of the glandular complex of the neck showed no alterations in any salivary glands, at the behest of the owner, we only performed the marsupialization. Initially, the plan was to surgically repair both ranulas. However, upon visualization of gelatinous material instead of expected saliva-filled cavity, the decision of stage approach following histopathological evaluation was made.Shar-pei dogs, more than other breeds, are predisposed to developing cutaneous mucinosis. In our case, skin biopsies were also performed, confirming the coexistence of both conditions.It is presently unknown whether the oral and cutaneous localizations could be related, but being cutaneous disease a probably genetic based disease , this waA positive correlation is reported between increased HA serum concentrations and cutaneous mucinosis in dogs Zanna, et al. . In the In conclusion, in case of sublingual swelling in Shar-pei dogs or in dogs with suspected cutaneous mucinosis, oral mucinosis should also be considered as a differential diagnosis. In our case, the surgical treatment for this type of pathology was considered curative with a positive prognosis.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.Ethical review and approval was not required for the animal study because the case report is a description of a clinical case. Written informed consent was obtained from the owners for the participation of their animals in this study.All authors listed have made a substantial, direct, and intellectual contribution to the work and approved it for publication.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.


Acute promyelocytic leukemia (APL) constitutes a serious hematological emergency necessitating rapid diagnosis and therapy to prevent lethal bleedings resulting from APL-induced thrombocytopenia and coagulopathy. Atypical manifestations of APL, such as extramedullary disease at first presentation, pose diagnostic challenges and delay the onset of appropriate therapy. Nevertheless, extramedullary manifestations of APL are mostly accompanied by blood count alterations pointing to an underlying hematological disease. In this report, we present the first case of APL bearing close resemblance to a metastasized laryngeal carcinoma with normal blood counts and absent coagulopathy.via magnetic-resonance imaging (MRI) and positron emission tomography–computed tomography (PET/CT) with F-18-fluordeoxyglucose (FDG) showed a large hypermetabolic tumor in the right piriform sinus and tracer uptake in adjacent lymph nodes, highly suspicious of metastasized laryngeal carcinoma. Surprisingly the histological examination revealed an extramedullary manifestation of acute promyelocytic leukemia. Remarkably, blood counts and coagulation parameters were normal. Moreover, no clinical signs of hemorrhage were found. PML-RARA fusion was detected in both laryngeal mass and bone marrow. After diagnosis of APL, ATRA-based chemotherapy was initiated resulting in complete remission of all APL manifestations.A 67-year-old man with a previous history of smoking was admitted to our hospital with progressive hoarseness of voice, odynophagia, dysphagia and exertional dyspnea. Laryngoscopy revealed a fixed right hemi larynx with an immobile right vocal fold. Imaging of the neck This is the first case report of APL initially presenting as laryngeal chloroma. Additionally, we performed a comprehensive literature review of previously published extramedullary APL manifestations. In aggregate, a normal blood count at first presentation constitutes an extremely rare finding in patients initially presenting with extramedullary APL manifestations. PML-RARA fusion gene originating from a reciprocal translocation connecting the long arms of chromosomes 15 and 17 [t] constitutes the signature genetic alteration of APL (PML-RARA without an underlying translocation were reported (RARA gene located on chromosome 17 resulting in a differentiation arrest at the promyelocytic stage (Formerly classified as AML M3 according to the French-American-British (FAB) classification , acute ptq4.1;q21.1The initial diagnosis of APL, which accounts for 5-10% of all newly diagnosed AML cases, poses a critical hematological emergency . The higAtypical manifestations of APL compound swift diagnosis and delay the initiation of induction chemotherapy. Recently, a rare case of extensive extramedullary APL lesions with infiltration of the clivus, mandible and maxilla was published. Nevertheless, pancytopenia, coagulation alterations and characteristic bone marrow findings enabled early diagnosis of APL .We present another atypical case of APL bearing close resemblance to a locally metastasized laryngeal carcinoma. In this case, however, normal blood counts, normal coagulation parameters and a discontinuous bone marrow involvement causing false negative bone marrow aspiration results additionally complicated APL-diagnosis. The aim of this case report is to highlight unprecedented obstacles to swift APL diagnosis.PML-RARA fusion transcripts were found. The cytogenetical analysis yielded a normal male karyotype with no apparent alterations. Notably, no translocation t could be detected via fluorescence in situ hybridization (FISH). Due to the low signal intensity of PML-RARA, additional molecular genetic examinations of peripheral blood and skin chloromas were performed. The PML-RARA fusion was detected by nested PCR in both blood and skin samples, confirming the diagnosis of APL with cryptic PML-RARA rearrangement.A 67-year-old man with a 2-month history of progressive hoarseness of voice, odynophagia, dysphagia, and exertional dyspnea presented to our hospital. In addition, he complained about a new growing skin lesion on his back. The patient had a previous history of smoking, but he reported ongoing abstinence from nicotine for 17 years. In the clinical examination, several subcutaneous nodules located on the right chest, on the head and on the back were found. Laryngoscopy revealed a fixed right hemilarynx with an immobile right vocal fold and a normal left vocal fold. Magnetic-resonance imaging (MRI) of the neck showed a large mass protruding into the right hemilarynx  and daunorubicin . Having corroborated the diagnosis of APL based on the evidence of risk APL . At the PML-RARA is a vital precondition for the diagnosis APL, the signature translocation t is found in more than 90% of all APL cases, but it does not constitute an essential diagnostic criterion  before  did further delay the diagnosis of APL, as the weak intensity of the PML-RARA signal required a second confirmatory PCR. Usually, a protracted onset of chemotherapy diminishes the survival of patients with APL by raising the chances for serious hemorrhage. Fortunately, in this case, the initial diagnosis of APL was complicated by absent hematological abnormalities reducing the jeopardy for acute incidents of serious bleeding. Due to the pronounced extramedullary leukemia formation, the patient was assigned to the high-risk group of APL patients, albeit all blood counts were within normal range. This procedure is in line with a previous case report about a patient exhibiting extensive intracranial infiltration of APL, who was subjected to intensified therapy despite low risk disease according to Sanz risk score calculated from peripheral leukocyte and thrombocyte count  before Table 1.cation t1;17 is focation t1;17 is fote count , 31. Furte count , 33. Accte count .In aggregate, we report the first case of APL initially manifesting with a laryngeal chloroma, normal blood counts and absent coagulopathy. Finally, we present a striking discrepancy between results from examination of bone marrow aspirate and histological analysis. In light of the possible fatal repercussions of delayed APL diagnosis, we want to enlarge the repository of atypical APL manifestations to enable swift APL diagnosis even in more complicated cases.The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.DCH, FL, WH, AR and DH treated the patient. IE, KM and DiH performed all imaging (radiology/nuclear medicine). KU analyzed all histopathological examinations. DCH and DH wrote the manuscript. All the authors revised the manuscript critically, approved the final manuscript, and agreed to be accountable for all aspects of the manuscript.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.


A meconium pseudocyst is formed following meconium peritonitis. At present, antenatal diagnosis and planned management of meconium pseudocyst have reduced the mortality rate significantly. We presented a case of a neonate with abdominal distension and non-passage of meconium who experienced respiratory distress and was taken for exploratory laparotomy at a tertiary care center due to suspected bowel perforation. The neonate was diagnosed with a meconium pseudocyst intraoperatively as maternal ultrasound and ultrasound of the abdomen of the neonate after birth failed to make a definitive diagnosis; even an X-ray abdomen did not reveal pathognomonic egg-shell calcification. An interesting aspect of this case is the mother’s complex obstetric history, which compelled us to conjecture whether it was possible to predict the chances of meconium peritonitis and take steps to prevent it. It must be noted that, despite rigorous research, the researchers could not find reliable literature co-relating the obstetric history of the mother with the formation of a meconium pseudocyst in the neonate.  Meconium pseudocyst formation is the consequence of the in-utero intestinal obstruction or ischemia of the bowel leading to perforation. Leaked meconium is sterile but causes a chemical irritation of the peritoneum. To control the leaked meconium, a pseudocyst is formed. The affected infant may experience non-passage of meconium, abdominal distension, guarding, or rigidity -3.It has been possible to make an antenatal diagnosis of a meconium pseudocyst can through antenatal ultrasound scans of the mother since 1980. This procedure has reduced the mortality rate from 50% to 11%; in addition to diagnosis, it has also assisted in deciding the need for post-natal surgery in 50% of the patients with persistent ascites and signs of obstruction such as dilated bowel loops . A more A 35-year-old Indian female with a history of gravida 3 Para 1 live 1 abortion 1 came to the emergency department at a rural hospital in Central India following pre-term labor pain and gave birth to a 2 kg female neonate via lower-segment cesarean section. On further detailed inquiry, her history of ovulation induction followed by twin gestation was revealed. Since the woman already had birthed a male child with Duchenne muscular dystrophy, she underwent selective fetoreduction at the gestational age of 14 weeks. The scan at the gestational age of 21 weeks was suggestive of marginal placenta previa grade 1, and she was diagnosed with hypothyroidism at 34 weeks of pregnancy. The mother underwent multiple prenatal ultrasonography, but none were suggestive of fetal bowel obstruction. Prenatal ultrasound was done on admission at the gestation age of 34 weeks and six days and reported polyhydramnios with grade 3 placenta previa with the possibility of placenta accreta and small bowel obstruction.Immediately after birth, the female neonate was admitted to the neonatology department of the hospital and was referred to a pediatric surgeon in view of abdominal distension and non-passage of meconium since birth. On examination, the neonate had abdominal distension with erythematous skin over the abdomen, which increased over four hours after birth. The infant feeding tube was also not stained with meconium on per-rectal examination. The nasogastric tube had about 15 ml of bilious aspirate. A roentgenogram (X-ray) of the abdomen in an erect position revealed a large mass lesion on the right side of the abdomen displacing bowel loops on the left side (Figure Exploratory laparotomy was done through a right transverse supraumbilical incision. As soon as the peritoneum was incised, about 200 ml of watery, coffee-ground-colored fluid drained, as the pseudocyst wall was densely adhesive to the abdominal wall and bowel loops. On careful and gentle separation of the pseudocyst wall, the bowel was visible. The bowel was inspected along the length and found to be atretic at the terminal ileum. A short length of the ileum distal to the atretic segment measuring approximately 2 cm along with the cecum and appendix was excised, and a double barrel ileostomy was created (Figure The neonate did not cry immediately after birth and even after 15 seconds of positive pressure ventilation and was, hence, intubated immediately and shifted to the neonatal intensive care unit. She was started on injectable antibiotics ampicillin and gentamycin along with other supportive care. Post-operatively, she was unable to maintain adequate saturation and blood pressure, and hence, despite all efforts, including inotropic support, the neonate patient succumbed to her illness on postoperative day one.  A meconium pseudocyst is formed following a meconium leak, that is, intrauterine bowel perforation leading to enteric secretions that come in contact with the peritoneal cavity causing a fibrous reaction. It is essentially a fibrous wall formation around the leaked meconium, which is majorly following antenatal bowel obstruction and perforation secondary to meconium ileus, bowel atresia, intussusceptions, Meckel’s diverticulum, Hirschsprung’s disease, idiopathic cause, and sigmoid volvulus It may present as a palpable abdominal lump, progressive abdominal distension, respiratory distress, or sepsis. According to the meta-analysis conducted by Shinar et al.  on fetalSurgery is the standard for the treatment of meconium pseudocysts; surgical procedures consist of drainage procedures and stoma formations, followed by ostomy closure in a second sitting. Notably, a recent retrospective study with 38 patients demonstrated improved survival with primary anastomosis, except for low birth weight babies . The intBased on the case presented, it should be noted that since there was a long complicated maternal history, including fetal reduction, hypothyroidism, and ovulation induction, the etiology for the intestinal perforation causing meconium pseudocyst formation could not be determined. However, one can wonder if the perforation was iatrogenic or spontaneous. Does gestational hypothyroidism in mothers affect bowel growth and development? There is not much evidence supporting and proving all the above claims. Reporting more such cases in detail would help us better understand how to prevent and manage meconium pseudocysts/peritonitis to achieve increased survival among neonates.


Chlamydia trachomatis. The underlying cause of her small bowel obstruction could be due to pelvic inflammatory disease, Fitz-Hugh-Curtis syndrome, or endosalpingiosis. We aimed to create awareness amongst readers that small bowel obstruction in young female patients with no prior abdominal surgery is possible and often difficult to diagnose immediately.A 19-year-old female has multiple presentations to emergency department with recurrent abdominal pain. During her third presentation, the radiological features were suggestive of high-grade small bowel obstruction in a virgin abdomen. A diagnostic laparoscopy has been performed. The intraoperative findings include a band adhesion between omentum and small bowel mesentery, and perihepatic adhesions consistent with Fitz-Hugh-Curtis syndrome. The histopathology from a biopsy of the macular lesions of the abdominal wall showed endosalpingiosis. The postoperative high vaginal swab was positive for  A small bowel obstruction in a virgin abdomen is rare and has to be treated with caution. The potential underlying causes include but not limited to internal hernia, congenital adhesions, endometriosis, and pelvic inflammatory disease. In this case report, we will discuss about the presentation of a young female patient to the hospital, the investigations involved and the management plans. We will also discuss in detailed about Fitz-Hugh-Curtis syndrome, endosalpingiosis, and pelvic inflammatory disease in the context of small bowel obstruction.In this report, we describe a case of a 19-year-old female who presented to a regional hospital emergency department with increasing colicky abdominal pain, describing at least seven days of obstipation, intolerance to oral intake, and multiple large amounts of vomits. This was her third presentation within nine days. On this occasion, the general surgery service was consulted, and the computed tomography of the abdomen and pelvis with intravenous contrast was performed. Imaging demonstrated a high-grade small bowel obstruction (SBO) with a transition point in the pelvis.The patient's medical history included primary dysmenorrhea. She tried oral contraceptive pills and Implanon but tolerated them poorly to their effect on mood. A recent gynaecological endocrine panel was unremarkable. She was on the waitlist for a diagnostic laparoscopy to investigate for suspected endometriosis. The previous sexually transmitted infection (STI) screening was reported to be negative. She had not had any abdominal operations and did not take any regular medications.After gastric decompression and intravenous fluid resuscitation, the patient was transferred to a tertiary referral hospital and proceeded to diagnostic laparoscopy. Operative findings were of dilated loops of small bowel with a band adhesion caused by the omentum adhered to small bowel mesentery . There wChlamydia trachomatis. Unexpectedly, the intraoperative histopathology did not identify endometriosis but rather endosalpingiosis. It is a less well-studied entity and has not been associated with intestinal obstruction in published literature. The patient progressed to a complete diet and was discharged on the second postoperative day with gynaecological follow up.Postoperative gynaecological review elucidated the further history of deep dyspareunia. The high vaginal swab was positive for The small bowel obstruction (SBO) in a virgin abdomen is rare and may be caused by congenital adhesions, small bowel tumours, internal hernia, or gallstone ileus . In thisEndosalpingiosis is described as ectopic glands of fallopian tube-type ciliated epithelium. It may occur in female reproductive and other visceral or retroperitoneal organs, either in isolation or concurrently with endometriosis or endocervicosis. The clinical features of endosalpingiosis are not well described. It may be associated with pelvic pain, infertility, and urinary symptoms. Literature reports a histologic relationship between endosalpingiosis and pelvic serous neoplasms, whether as a risk factor or disease process is unclear . HistoloChlamydia trachomatis with pelvic inflammatory adhesions and Fitz-Hugh-Curtis syndrome. The numerous hospital presentations with right upper quadrant pain may have been related to the liver capsule inflammation. SBO caused by PID is rare, and there are only 13 cases reported in the English literature to our knowledge  described in the literature [The other diagnosed concurrent pathology was nowledge . The majn et al.  bears thterature .This is the first reported case of SBO with histological findings of endosalpingiosis. It is however difficult to prove if it was the true causative aetiology for her presentation. Another possibility is that her SBO was due to endometriosis –8.. WhilIn this case report, we have identified a young, fertile female patient with recurrent presentations to the hospital with abdominal pain. She had an underlying SBO with no prior abdominal surgery. The cause of her SBO could be related to the underlying clinical pathologies as discussed above. PID and endometriosis may be important to be identified early to prevent future SBO.


Background: SO (Struma ovarii) is a rare form of ovarian teratoma which originates from ovarian dermoid cysts. Due to the rarity of this disease, relevant studies might not be sufficiently documented, especially cases with hyperthyroidism and multiple metastases.Case Presentation: A 40-year-old female patient was admitted to our hospital due to management of early pregnancy along with a recurrent abdominal and pelvic mass. Contrast-enhanced CT images showed an irregular mass (10.7 × 8.6 × 12.8 cm) located in the right side from the hypogastrium to the pelvic cavity and another mass (3.8 × 3.7 cm) in the liver. Laboratory examination showed that CA125 (Carbohydrate Antigen-125) was 118.10 U/mL, Tg (thyroglobulin) was >300 ng/ml, FT4 (free thyroxine) was 22.11 pmol/L, and TSH (thyroid-stimulating hormone) was <0.004 mIU/L. She subsequently underwent liver mass dissection, omentectomy, tumor dissection, peritoneal nodule resection, as well as rectal anterior wall nodule resection. The patient was diagnosed with malignant SO (papillary type) along with multiple metastases. Also, we conducted a literature review based on 290 SO cases from 257 articles.Conclusion: This study showed that malignant SO might be prone to relapse and metastasize (a metastatic rate of 52.94%) and therefore aggressive management might need to be recommended for malignant SO. Also, laparotomy might need to be recommended for large tumors that cannot be resected by laparoscopic surgery since these tumors might be prone to rupture and thus produce peritoneal implants. Furthermore, Graves’ disease might need to be considered in the differential diagnosis. SO (Struma ovarii), a rare form of ovarian teratoma which originates (more than 50%) from ovarian dermoid cysts consisting of thyroid tissue, accounts for about 1% of all ovarian neoplasms and about 2% of mature ovarian teratomas , 4. Due The differential diagnosis between malignant SO and benign SO varied largely over the years. Some researchers hypothesize that malignancy can only be diagnosed when the tumor shows definite invasion or metastases, while others diagnose malignancy on nuclear or histologic alterations . In mostA 30-year-old female with early pregnancy and a left ovarian mass was admitted to a local primary hospital, where she later received a laparoscopic mass dissection and induced abortion in January 2008. Postoperative histology suggested the presence of an inclusion cyst. In August 2008, the patient was readmitted to that hospital due to a right ovarian mass with the greatest dimension up to 10 cm. Then, the patient received another laparoscopic resection of the mass which ruptured in the pelvic cavity during the dissection. Postoperative histology confirmed that the mass was composed of SO . The patient did not have any follow-up until 5 years later when a new pelvic cystic mass (4.0 cm in greatest dimension) was discovered. However, she declined treatment. Then in April 2018, at the age of 40, the patient was admitted to the obstetrics and gynecology department of our hospital for management of another early pregnancy along with a recurrent abdominal and pelvic mass which had enlarged from 4.0 to 9.0 cm.During the physical examination, there were no remarkable findings, apart from a palpable mass in the right lower abdomen with no tenderness or rebound tenderness.Contrast-enhanced CT (computed tomography) images of the whole abdomen and pelvic cavity showed an irregular mass (10.7 × 8.6 × 12.8 cm) located in the right side from the hypogastrium to the pelvic cavity containing both solid and cystic components with regional irregular calcifications. The mass was surrounded by multiple enhancing lesions and low-density nodules of different sizes with the largest one up to 4.3 cm in diameter . In addiLaparoscopy showed multiple light red nodules which were each about 1.0 cm in diameter in the greater omentum and the right side of the peritoneum, one large exophytic mass (5.0 cm in diameter) with a grey surface in the S6 segment of the liver (without invasion of the liver parenchyma), as well as a large polycystic mass in the right adnexal region with a high-surface-tension and a complete capsule. No ascites had formed in the patient’s abdomen. A diagnostic resection of the peritoneal nodules was performed using an ultrasonic scalpel and tissues were immediately sent for rapid pathological examination. It showed that components of thyroid tissue could be found in the nodules and thus they were considered to be metastatic lesions that might arise from the SO. Subsequently, a liver mass dissection and omentectomy were performed in sequence. Laparoscopic instruments were then withdrawn. Laparotomy was immediately performed. During the laparotomy, total tumor dissection, unilateral salpingo-oophorectomy, peritoneal nodule resection, as well as rectal anterior wall nodule resection were performed. Due to the patient’s strong will of preserving her fertility, the uterus and the left ovary were preserved.Pathologic examination after the surgery indicated that the thyroid component could be found in the tissues removed from the right ovary. Hematoxylin-eosin (HE) staining showed that there was a presence of papillae. Irregular, enlarged, ground glass, and overlapping nuclei with grooves and pseudoinclusions could also be found after staining. The tumor was entirely composed of the above component and there was no evidence of benign SO. Immunohistochemically, Galectin-3, CK19 (cytokeratin-19), TTF-1 (thyroid transcription factor-1), and Ki-67 had positive expressions . TissuesA laboratory test was performed at 1-month follow-up after the surgery and it showed a normal level of FT4 (11.28 pmol/L), TSH (2.518 mIU/L), and Tg  patient data and these cases were excluded from further analyses to ensure the accuracy of this study.A PubMed literature search of SO case report was performed, with the use of the following terms: “struma ovarii,” “malignant struma ovarii,” and “benign struma ovarii.” A total of 484 articles were retrieved in the search. After selection, 257 eligible articles containing 290 eligible cases were reviewed . The search criteria were shown in p value = 0.292, p value = 0.410). Also, no significant statistical difference was found in tumor size between these two groups . For clinical symptoms, no significant statistical differences were found whether in abdominal discomfort, abdominal genital bleeding, or urinary compressive symptom between malignant SO and benign SO , but the occurrence of hyperthyroidism was higher for benign SO than that of malignant SO . For serum tumor marker CA125, more positive cases were observed in benign SO with a higher serum level than those of malignant SO . Malignant SO had a higher rate of metastasis than that of benign SO and there was a significant statistical difference .As shown in Mature teratomas are composed of tissues from ectodermal, mesodermal, and endodermal origins . SO, oriK Devaney et al. as follows . The study of Wei, S et al., also showed that papillary type was the commonest well-differentiated neoplasm arising in SO . This clClinical characteristics of SO may include lower abdominal discomfort, abnormal vaginal bleeding and some of those cases may also present ascites, hydrothorax, or hyperthyroidism , 23. Ourp value = 0.001). We suspected that malignant SO is often poorly differentiated, and thus it rarely develops functioning thyroid tissue. In our case, despite the malignancy, laboratory examination also showed hyperthyroidism. Since no abnormality was detected in ultrasonography when examining the thyroid gland and both FT4 and TSH levels became normal after the surgery, the hyperthyroidism was likely caused by the hyperfunctioning SO .This study showed that the positivity rate of CA125 was about 57.27% in all tumors . This result was relatively consistent with previous studies which suggested that the positive value of CA125 was around 60% for all ovarian cancers , 29. ThiSerum Tg may also be a useful indicator in terms of the diagnosis and monitoring of SO. Several studies suggested that the elevated Tg concentration was a useful indicator in the diagnosis and Tg level could be used for detecting the recurrence of SO for patients who received total thyroidectomy . Our casp value = 0.001). The study of Marti JL, et al. also suggested that thyroidectomy with RAI therapy is reasonable when extra-ovarian spread, metastases, or synchronous primary thyroid gland cancer are present  with mixed density and irregular enhancement in the left adnexal region 6 months after the laparotomy. Although the histology could not be determined because the patient refused any other treatment, we suspected that this might be another recurrent SO. This case report and literature review might be helpful to some extent in the knowledge of this disease.123I scan was not performed due to the concerns of avoiding overdiagnosis. Second, analytic results of the literature review only represent cases that have been published to date and they might change to some extent in the future when more cases are added.There were two limitations in this study. First, hyperthyroidism seen in this patient was diagnosed based on the fact that thyroid hormone and TSH levels normalized after resection. However, a This case report showed that the patient presented with SO repeatedly, which indicated that malignant SO might be prone to relapse. Besides, our literature review also showed that malignant SO often has a high metastatic rate (52.94%). These results suggested that aggressive management including total hysterectomy, bilateral salpingo-oophorectomy, and total thyroidectomy followed by postoperative RAI treatment might need to be recommended for malignant SO. Also, laparotomy might need to be recommended for large tumors that can not be resected by laparoscopic surgery since these tumors might be prone to rupture and thus produce peritoneal implants. Furthermore, a differential diagnosis of other causes of hyperthyroidism might need to be considered.At 1 month follow-up after the laparotomy, the patient and her family were satisfied with the treatment effect.


Soft tissue sarcomas arise infrequently and make up less than 1% of all cancers. An undifferentiated pleomorphic sarcoma (UPS), formerly known as a malignant fibrous histiocytoma, is a malignant subtype of soft tissue sarcoma identified by a lack of specific immunohistochemical markers for lineage differentiation. These tumors are aggressive and enlarge rapidly with an increased risk of metastasis, thus prompt diagnosis, treatment, and post-resection surveillance are imperative. This case report demonstrates an incidental finding of a large undifferentiated pleomorphic sarcoma of the posterior thigh in an 86-year-old male initially evaluated with a bedside ultrasound. Soft tissue sarcomas arise infrequently and make up less than 1% of all cancers. An undifferentiated pleomorphic sarcoma (UPS), formerly known as a malignant fibrous histiocytoma and regrouped by the World Health Organization in 2002, is a malignant subtype of soft tissue sarcoma . The neoRisk factors for UPS include previous radiotherapy and older age . MoreoveThe patient is an 86-year-old male with no significant past medical history who initially presented to the emergency department for pain and swelling of the right posterior thigh for the past one week. Family members noted the patient has had the mass for years, but over the past few days it had increased in size, and therefore, the patient was brought in to be evaluated in the emergency department. The patient’s physical examination was unremarkable except for moderate swelling and tenderness of the right posterior thigh with no distal erythema or palpable cord. In the emergency department, a bedside ultrasound Figure  of the rSubsequent CT imaging of the right lower extremity with IV contrast Figure  revealedUPS, also known as malignant fibrous histiocytoma, is classified as a high-grade pleomorphic malignant tumor that lacks specific immunohistochemical markers for lineage differentiation . UPS is The standard of care for UPS includes an en bloc surgical resection with microscopically negative margins followed by serial monitoring by CT or MRI for surveillance of recurrence or late pulmonary metastasis . The rolUPS is rare and delay to diagnosis is common as the neoplasms are not typically included in the physician’s differential diagnosis. Moreover, because UPS usually presents as asymptomatic, rapidly growing cutaneous or subcutaneous nodules without superficial skin abnormalities, patients often do not seek immediate care . Our patFurthermore, the diagnostic evaluation of UPS varies depending on the location of the tumor. MRI is recommended for initial imaging of soft tissue masses in the head/neck, trunk, and extremities, while CT is recommended for imaging of retroperitoneal and visceral masses . HoweverTo date, evidence is lacking to recommend sentinel lymph node biopsy or positron emission tomography for the initial staging of soft tissue sarcoma. Due to its high diagnostic accuracy, a core needle biopsy is preferred over an open incisional biopsy for the initial evaluation of UPS. The latter should be performed by an experienced surgeon considering the future resection incision, in the case of our patient, and to be sure to minimize dissection and hemorrhage risk . Due to Soft tissue sarcomas arise infrequently and make up less than 1% of all cancers; however, they should be included in the differential diagnosis when a patient presents with a rapidly enlarging, painless mass in the head/neck, trunk, extremities, or retroperitoneum. Sarcomas enlarge rapidly, as evident by our patient with a 16.0 × 8.1 × 9.5 cm tumor at presentation. It is imperative to prioritize prompt diagnosis as the risk of metastasis increases as the tumor grows in size and/or invades local structures; bedside ultrasound may serve as a fast, non-invasive, and inexpensive initial diagnostic test. MRI and CT are currently the preferred imaging choices, in conjunction with core needle biopsy, to establish the diagnosis. At present, standard treatment consists of en bloc surgical resection with microscopically negative margins. Serial surveillance with MRI or CT should be conducted after surgical resection to monitor for recurrence or metastasis.


Cardiac paragangliomas (PGLs) are clinically rare, with hypertension and metabolic changes as the main symptoms. The tumor is highly related to gene mutation, and surgery is presently the effective treatment. Medical history and clinical manifestations of the patient, routine laboratory examinations and imaging examinations, and pathological examination can help the final diagnosis.The present study presents a 31-year-old male patient with a left atrial mass. The initial symptom was cough. Cardiac enlargement was found during the chest X-ray. The follow-up imaging examination revealed a left atrial occupying lesion, and the possibility of malignant occupying lesions was not ruled out. The patient underwent surgical resection of the mass. The final pathological result revealed paraganglioma. The thoracic computed tomography review two months after the operation revealed that the original occupying lesion disappeared, and no new lesion was found.Pheochromocytomas and paragangliomas (PPGLs) are a kind of neuroendocrine tumors. PPGLs can cause secondary hypertension, and lead to a series of clinical syndromes, including myocardial injury, metabolic changes, and so on. The occurrence of PPGIs is related to gene mutation. Biochemical detection, imaging examination, and genetic testing can help diagnose. The tumor should be surgically removed as soon as possible after the diagnosis. As a functional tumor, PPGLs should be fully prepared before surgery to avoid anesthesia and huge fluctuations in blood pressure during and after surgery, or the occurrence of fatal hypertensive crisis and intractable hypotension after tumor resection. Adequate preoperative preparation directly affects the prognosis of patients after surgery. Therefore, multidisciplinary cooperation before, during, and after the operation is extremely important. Pheochromocytomas and paragangliomas (PPGLs) are a class of neuroendocrine tumors that originate in the adrenal medulla and extra-adrenal sympathetic chain, respectively. PPGLs are capable of the paroxysmal or persistent secretion of catecholamines, causing secondary hypertension and contributing to a range of clinical syndromes, including myocardial damage, metabolic alterations, and so on . PPGLs aA 31-year-old male was referred to our hospital with a tumor in the left atrium (LA). Half a month before coming to our hospital, the patient went to a local hospital for treatment due to an intermittent cough. The chest X-ray revealed no obvious inflammation in the lungs, but the heart was dilated. The transthoracic echocardiography indicated a left atrial occupying lesion and a large amount of pericardial effusion. Pericardiocentesis was performed, and 600 mL of bloody pericardial effusion was removed. Then, the patient visited our hospital for further treatment.The patient had no family history related to tumors or environmental exposure histories, such as chemical substances and high radiation. The patient lost 5 kg of weight since its onset. Routine laboratory examinations were performed after admission Table . The traAfter a series of examinations, the investigators decided to surgically remove the tumor. During the preoperative anesthesia, the blood pressure remained at a high level, despite the drug intervention. After the general anesthesia, the investigators performed a median incision on the chest and exposed the heart position. After establishing the cardiopulmonary bypass, the right atrium and atrial septum were incised, and it could be observed that the intima of the LA was smooth and intact. Then, the location of the tumor was explored. It was observed that the tumor was located within the wall of the LA, and adhered to the right pulmonary artery. The tumor was carefully separated from the heart adhesion to completely remove the tumor. Finally, left atrial reconstruction was performed with commoditized bovine pericardium. It is noteworthy that during the surgery, regardless of whenever the tumor was touched, the patient’s blood pressure transiently raised. The tumor was approximately 6.5 cm in length. The removed tumor was submitted for pathological analysis, and the final result was suggestive of paraganglioma Fig. . The patPheochromocytomas (PCCs) are located in the adrenal glands, while paragangliomas (PGLs) are located along the paraspinal sympathetic chain and are classified as hormonally active or inactive, and both of which are jointly known as PPGLs , 5. The Although rare, PPGLs are the most heritable cancer type, with 40% of patients carrying predisposing germline mutations . In 2017The main signs and symptoms of excess circulating catecholamines from PPGL are headache, palpitations, sweating, pallor, nausea, constipation, flushing, weight loss, fatigue, anxiety, sustained or paroxysmal hypertension, orthostatic hypotension, fever, and hyperglycemia , 14. TheThe present case provides valuable experience for the treatment of similar patients in the future. After the diagnosis, the tumor should be removed by surgery as early as possible. Surgery can improve the symptoms, reduce hormone secretion, prevent associated complications, and improve the outcome of subsequent treatments. However, crushing the tumor during surgery can lead to the massive release of catecholamines, which may cause a hypertensive crisis, arrhythmia, stroke, etc. Furthermore, postoperatively, the resulting dramatic reduction in catecholamines may cause peripheral vasodilatation, causing persistent hypotension, and even death. Therefore, adrenergic α-blockers can be routinely used preoperatively, and β-blockers can be added if the patient develops tachycardia . The useAs a consequence, during the patient’s hospitalization, multidisciplinary consultation should be strengthened, including the Endocrine Department, Intensive Medicine and Anesthesiology Department, and other disciplines, in order to ensure patient safety maximization. Furthermore, postoperative long-term follow-ups should be conducted.


Non‐secretory multiple myeloma (NSMM) is an extremely rare variant of multiple myeloma (MM) and accounts for a maximum of 5% of all myeloma cases. This variant of MM usually represents a diagnostic challenge to the clinician because of the absence of detectable monoclonal immunoglobulin on serum or urine electrophoresis. We present the case of a 34‐year‐old Caucasian male who presented to the emergency department with pain in the lumbar area secondary to a fall and who was eventually diagnosed with non‐secretory multiple myeloma after the radiologist initially pointed out a discrete “salt and pepper” infiltration of the spine seen on magnetic resonance imaging (MRI) although the spine computed tomography (CT) performed initially showed no suspicious lesions for malignancy. The final diagnosis was obtained after a positive bone marrow biopsy together with the presence of malignant lesions seen on the spine MRI. This case points out the importance of different bone marrow involvement patterns seen on MRI and other useful sequences the radiologist could use to better discriminate between normal marrow reconversion and malignant infiltration. We describe this case report to raise awareness among medical practitioners and radiologists of the extreme importance of subtle bone marrow infiltration patterns seen on MRI, which could help diagnose multiple myeloma at an early stage. Non‐secretory multiple myeloma (NSMM) on the contrary is a rare variant of MM which accounts for a maximum of 5% of all myeloma cases and is even much rarer in young people.Both entities have the same clinical and imaging features. However, in the case of NSSM, the plasma cells fail to secrete an immunoglobulin, and therefore, both the serum and urine electrophoresis are normal.The use of MRI in the diagnosis and staging of MM has increased within the last decades, MRI being more sensitive than computed tomography and X‐rays in the detection of focal MM lesions, as well as subtle medullary infiltration.We present the case of a patient with non‐secretory multiple myeloma that was diagnosed at an early stage, before end‐organ damage, thanks to the extreme sensitivity of MRI in depicting marrow infiltration, changes that in numerous instances are visible before any clinical or serological abnormalities are observed.2A 34‐year‐old male patient presented to the Emergency Department of the Regional Hospital, complaining of lumbar back pain after a fall from a 2‐m height. The lumbar spine CT was normal Figure , demonstAfter extensive laboratory results, nothing but discrete anemia was noted; there was no detectable monoclonal immunoglobulin on serum or urine electrophoresis Table .The patient continued to complain of worsening lumbar pain for the next 2 weeks, and another MRI was performed; this time the presence of a new nodular infiltration of the spine at the level of the L1 vertebral body  chemotherapy protocol for up to 8 cycles, every 21 days. The treatment was well tolerated with minimal side effects, mainly fatigue, headaches, and nausea. All the side effects responded well to symptomatic drugs. The patient is currently in complete remission with normal laboratory results except for slightly elevated AST (44 UI/L) and ALT (56 UI/L) levels which were considered secondary to therapeutic hepatotoxicity.3NSMM is a rare type of MM and is defined by the absence of detectable M protein in serum and urine. Two different subtypes of NSMM have been described. In the first subtype, the plasma cells produce immunoglobulins but are unable to release them out of the cell, probably due to decreased permeability, deficiency, or damage of intracellular light chains. This variant of NSMM is known as the “producer” type. The second form is the “non‐producer” type, in which immunoglobulin cannot be produced by plasma cells.The diagnosis of multiple myeloma is based on the presence of bone marrow plasmacytosis of >10%, monoclonal protein in serum and/or urine, as well as myeloma‐related organ dysfunction (CRAB criteria).Due to the absence of M protein in serum and urine, our patient did not meet the criteria for the diagnosis of MM but was labeled as an NSMM after the second MRI scan revealed a new bone lesion at the level of the L1 vertebral body, several other enlarged lesions (between 5 and 10 mm) within the “salt and pepper” marrow infiltration and confirmed by a positive bone marrow biopsy for plasmacytosis.This case emphasizes the importance of modern imaging (MRI) in the diagnosis of a rare form of multiple myeloma,Computed tomography (CT) is a sensitive method for the diagnosis of lytic lesions, but it is well known that patients with multiple myeloma present bone marrow infiltration before any lytic lesions can be seen on a CT scan,Modern imaging methods like MRI detect discrete marrow infiltration patterns before lytic lesions are detected on computed tomography and sometimes before the onset of any symptoms.On MR imaging, five different patterns of bone marrow infiltration in multiple myeloma were described. These patterns include a normal‐appearing marrow, focal infiltration, diffuse infiltration, and “salt‐and‐pepper”; the fifth pattern is a combination of focal and diffuse infiltrationThe “salt and pepper” infiltration of the bone marrow is seen in only about 3% of MM patients,If in doubt, chemical‐shift imaging is a fast and easy way to discriminate between normal bone marrow and malignant infiltration, and therefore, it should be included in routine MRI examinations of patients with suspected marrow infiltration.Another fast MRI technique to help differentiate malignant marrow lesions from benign marrow reconversion is the “functional” diffusion‐weighted (DWI) sequence and the corresponding apparent diffusion coefficient(ADC). ADC values in active myeloma are significantly higher than normal red or yellow marrow,In the case presented, the first spine MRI scan was performed in a private imaging clinic with a routine scanning protocol that included only T1, T2, and T2 fat saturation sequences; that is why the radiology report only pointed out the discrete “salt and pepper” pattern without a certain diagnosis of multiple myeloma infiltration. If further scan sequences like chemical shift imaging or DWI were performed, the radiologist's report would have been certainly more precise and the bone biopsy requested earlier.4Multiple myeloma (MM) is a plasma cell dyscrasia associated with the clonal proliferation of a defective plasma clone. Non‐secretory multiple myeloma (NSMM) is a rare variant of MM and accounts for less than 5% of all MM cases and often poses diagnostic difficulties. We present the case of a patient with NSMM who was diagnosed after a series of 2 spinal MRIs which showed bone marrow infiltration suggestive of MM . This case emphasis the need for different MRI sequences, to better discriminate between normal marrow reconversion and malignant infiltration.DC was involved in literature research and manuscript writing. HI was involved in critical feedback and manuscript revisions.No funding was received.The authors declare that there is no conflict of interest.Written informed consent was obtained from the patient for the publication of this case report.


Pulmonary sclerosing pneumocytoma, formally named pulmonary sclerosing hemangioma, is a rare benign tumor with malignant potential often identified as solid pulmonary nodules. Sarcoidosis is an inflammatory, multisystemic disease of unknown cause with a wide range of clinical manifestations. The disorder is characterized by the formation of noncaseating granulomas in virtually any organ in the body. We present a case of a patient presenting with fever, weight loss, and respiratory symptoms found to have both a sclerosing pneumocytoma and pulmonary sarcoidosis. A diagnosis was made following the lobectomy. The patient was followed for two years with stable lymphadenopathy while remaining asymptomatic. First described by Liebow in 1956, pulmonary sclerosing pneumocytoma, formally named pulmonary sclerosing hemangioma, is a rare benign tumor with malignant potential ,3. PneumSarcoidosis is an inflammatory, multisystemic disease of unknown cause with a wide range of clinical manifestations believed to have been first described by Dr. Jonathan Hutchinson in 1877. The formation of noncaseating granulomas characterizes the disorder. It can affect virtually any organ in the body, predominantly the lungs, which is present in over 90% of patients . The incA 56-year-old woman presented to the emergency department with fever, 10-pound 4.54 kg) weight loss over three weeks, weakness, shortness of breath, and dry cough. She had a history of depression and was taking no regular medications. She had a four-pack-year history of smoking and quit at age 22. She had no symptoms of joint pain, rashes, uveitis, syncope, or palpitations. An initial chest x-ray was reported as normal. A thorax CT identified a right lower lobe nodule measuring 1.9 x 1.9 cm Figure , 3B and .54 kg we Given the diagnostic limitations of a small tissue sample from the needle biopsy, a definitive diagnosis could not be made; therefore, the patient underwent a right thoracotomy with right lower lobectomy and lymph node resection to allow examination of the entire lesion. On histopathology, a 2.3 cm well-demarcated nodule was identified Figure . The nodThe patient has been followed for two years with stable lymphadenopathy and has remained asymptomatic.The combination of a patient with both sclerosing pneumocytoma and pulmonary sarcoidosis is rare and has only been reported twice in the literature ,15. MostImportantly, clinicians should be aware that fine needle aspirations and frozen sections of pneumocytomas have often been misdiagnosed as lung adenocarcinoma . The assDespite being benign tumors, pneumocytomas can show progressive growth and can display patterns similar to those of active pulmonary tuberculosis on CT imaging ,19. TherPneumocytomas are difficult to diagnose on simple FNA and often require Immunohistochemistry staining. They have been shown to consist of moderately cellular, round epithelioid cells with clear cell features, columnar cells, and spindle cells. A papillary arrangement with prominent hyalinized fibrovascular cores is the most common architectural pattern, followed by flat sheets and acinar formations. Tumor cells demonstrate mild, focally moderate nuclear pleomorphism with prominent nucleoli, hyperchromasia, nuclear elongation, nuclear overlap, and occasional nuclear inclusions and grooves. In a study by Maleki et al., the background of samples consisted of foamy macrophages in nine cases, hemosiderin pigment in six cases, and lymphoid aggregates in three cases with no mitoses and/or necrosis . The staPneumocytomas are extremely rare benign lung tumors that often mimic lung malignancy in appearance and are occasionally mistaken for adenocarcinoma on biopsy. While pneumocytomas have rarely been reported to resemble active pulmonary tuberculosis, there does not appear to be an association with any other granulomatosis disease, with this only being the third reported case with sarcoidosis. While there does not appear to be any relationship between the two entities, this is an interesting case of a patient with two rare lung pathologies.