Dynamic ultrasonography for optimizing treatment position in superior mesenteric artery syndrome:Two case reports and review of literature

BACKGROUND Superior mesenteric artery(SMA)syndrome is a rare cause of duodenal obstruction by extrinsic compression between the SMA and the aorta(SMA-Ao).Although the left lateral recumbent position is considered effective in the treatment of SMA syndrome,individual variations in the optimal patient position have been noted.In this report,we present two elderly cases of SMA syndrome that exhibited rapid recovery due to ultrasonographic dynamic evaluation of the optimal position for each patient.CASE SUMMARY Case 1:A 90-year-old man with nausea and vomiting.Following diagnosis of SMA syndrome by computed tomography(CT),ultrasonography(US)revealed the SMA-Ao distance in the supine position(4 mm),which slightly improved in the lateral position(5.7–7.0 mm)without the passage of duodenal contents.However,in the sitting position,the SMA-Ao distance was increased to 15 mm accompanied by improved content passage.Additionally,US indicated enhanced passage upon abdominal massage on the right side.By day 2,the patient could eat comfortably with the optimal position and massage.Case 2:An 87-year-old woman with vomiting.After the diagnosis of SMA syndrome and aspiration pneumonia by CT,dynamic US confirmed the optimal position(SMA-Ao distance was improved to 7 mm in forward-bent position,whereas it remained at 5 mm in the supine position).By day 7 when her pneumonia recovered,she could eat with the optimal position.CONCLUSION The optimal position for SMA syndrome varies among individuals.Dynamic US appears to be a valuable tool in improving patient outcomes.

Managing adult-onset Still's disease in pregnancy:A case report

BACKGROUND Adult-onset Still’s disease(AOSD)is a rare systemic inflammatory disorder characterized by fever,arthritis,skin rash,and systemic symptoms.The etiology of AOSD is unknown;however,it is thought to be related to immune dysregulation.Although a rare disease,AOSD can significantly impact reproductive health,particularly during pregnancy.This case study assesses the implications of pregnancy in a patient with AOSD,as well as the potential for heredity of the disease.Neonatal hemophagocytic lympho-histiocytosis(HLH)is a rare and lifethreatening disorder characterized by hyperinflammation and uncontrolled activation of immune cells,leading to multiple organ dysfunction.This case report aimed to introduce neonatal HLH from a mother with AOSD.CASE SUMMARY This case study presents a 29-year-old female with AOSD who became pregnant and gave birth to a premature infant who was diagnosed with neonatal HLH.AOSD can significantly impact pregnancy and childbirth,as it may become more severe during pregnancy,with an increased risk of fetal loss and preterm birth.The management of AOSD during pregnancy involves the use of nonsteroidal anti-inflammatory drugs and glucocorticoids,as well as immunosuppressive agents in severe cases.However,the use of immunosuppressive agents during pregnancy may be associated with potential risks to the fetus.The hereditary implications of AOSD are unclear;however,available evidence suggests that genetic factors may play a role in the disease development.CONCLUSION AOSD can have significant implications for pregnancy and childbirth,including an increased risk of fetal loss and preterm birth.Neonatal HLH,a complication of AOSD in pregnancy,requires prompt diagnosis and management.Women with AOSD who are considering pregnancy should discuss their options with their healthcare provider and develop a management plan that addresses the potential risks to both mother and fetus.

New method of local adjuvant therapy with bicarbonate Ringer’s solution for tumoral calcinosis: A case report

BACKGROUND Tumoral calcinosis is a condition characterized by deposits of calcium phosphate crystals in extra-articular soft tissues,occurring in hemodialysis patients.Calcium phosphate crystals are mainly composed of hydroxyapatite,which is highly infilt-rative to tissues,thus making complete resection difficult.An adjuvant method to remove or resolve the residual crystals during the operation is necessary.CASE SUMMARY A bicarbonate Ringer’s solution with bicarbonate ions(28 mEq/L)was used as the adjuvant.After resecting calcium phosphate deposits of tumoral calcinosis as much as possible,while filling with the solution,residual calcium phosphate deposits at the pseudocyst wall can be gently scraped by fingers or gauze in the operative field.A 49-year-old female undergoing hemodialysis for 15 years had swelling with calcium deposition for 2 years in the shoulders,bilateral hip joints,and the right foot.A shoulder lesion was resected,but the calcification remained and early re-deposition was observed.Considering the difficulty of a complete rection,we devised a bicarbonate dissolution method and excised the foot lesion.After resection of the calcified material,the residual calcified material was washed away with bicarbonate Ringer’s solution.CONCLUSION The bicarbonate dissolution method is a new,simple,and effective treatment for tumoral calcinosis in hemodialysis patients.

Appendicitis combined with Meckel’s diverticulum obstruction, perforation, and inflammation in children: Three case reports

BACKGROUND Meckel’s diverticulum is a common congenital malformation of the small intestine,with the three most common complications being obstruction,per-foration,and inflammation.To date,only a few cases have been reported world-wide.In children,the clinical symptoms are similar to appendicitis.As most of the imaging features are nonspecific,the preoperative diagnosis is not precise.In addition,the clinical characteristics are highly similar to pediatric acute appendicitis,thus special attention is necessary to distinguish Meckel’s diver-ticulum from pediatric appendicitis.Patients with poor disease control should undergo laparoscopic exploration to avoid serious complications,including intestinal necrosis,intestinal perforation and gastrointestinal bleeding.CASE SUMMARY This report presents three cases of appendicitis in children combined with intestinal obstruction,which was caused by fibrous bands(ligaments)arising from the top part of Meckel's diverticulum,diverticular perforation,and diver-ticular inflammation.All three patients,aged 11-12 years,had acute appendicitis as their initial clinical presentation.All were treated by laparoscopic surgery with a favorable outcome.A complete dataset including clinical presentation,dia-gnostic imaging,surgical information,and histopathologic findings was also provided.CONCLUSION Preoperative diagnosis of Meckel’s diverticulum and its complications is challenging because its clinical signs and complications are similar to those of appendicitis in children.Laparoscopy combined with laparotomy is useful for diagnosis and treatment.

Giant Brunner's gland hyperplasia of the duodenum successfully resected en bloc by endoscopic mucosal resection: A case report

BACKGROUND Duodenal Brunner's gland hyperplasia(BGH)is a therapeutic target when complications such as bleeding or gastrointestinal obstruction occur or when malignancy cannot be ruled out.Herein,we present a case of large BGH treated with endoscopic mucosal resection(EMR).CASE SUMMARY An 83-year-old woman presented at our hospital with dizziness.Blood tests revealed severe anemia,esophagogastroduodenoscopy showed a 6.5 cm lesion protruding from the anterior wall of the duodenal bulb,and biopsy revealed the presence of glandular epithelium.Endoscopic ultrasonography(EUS)demonstr-ated relatively high echogenicity with a cystic component.The muscularis propria was slightly elevated at the base of the lesion.EMR was performed without complications.The formalin-fixed lesion size was 6 cm×3.5 cm×3 cm,showing nodular proliferation of non-dysplastic Brunner's glands compartmentalized by fibrous septa,confirming the diagnosis of BGH.Reports of EMR or hot snare polypectomy are rare for duodenal BGH>6 cm.In this case,the choice of EMR was made by obtaining information on the base of the lesion as well as on the internal characteristics through EUS.CONCLUSION Large duodenal lesions with good endoscopic maneuverability and no evident muscular layer involvement on EUS may be resectable via EMR.

Successful splenic artery embolization in a patient with Behçet’s syndrome-associated splenic rupture:A case report

BACKGROUND Splenic rupture associated with Behçet’s syndrome(BS)is extremely rare,and there is no consensus on its management.In this case report,a patient with BSassociated splenic rupture was successfully treated with splenic artery embolization(SAE)and had a good prognosis after the intervention.CASE SUMMARY The patient was admitted for pain in the left upper abdominal quadrant.He was diagnosed with splenic rupture.Multiple oral and genital aphthous ulcers were observed,and acne scars were found on his back.He had a 2-year history of BS diagnosis,with symptoms of oral and genital ulcers.At that time,he was treated with oral corticosteroids for 1 month,but the symptoms did not alleviate.He underwent SAE to treat the rupture.On the first day after SAE,the patient reported a complete resolution of abdominal pain and was discharged 5 d later.Three months after the intervention,a computed tomography examination showed that the splenic hematoma had formed a stable cystic effusion,suggesting a good prognosis.CONCLUSION SAE might be a good choice for BS-associated splenic rupture based on good surgical practice and material selection.

Link between mutations in ACVRL1 and PLA2G4A genes and chronic intestinal ulcers:A case report and review of literature

BACKGROUND Genetic factors of chronic intestinal ulcers are increasingly garnering attention.We present a case of chronic intestinal ulcers and bleeding associated with mu-tations of the activin A receptor type II-like 1(ACVRL1)and phospholipase A2 group IVA(PLA2G4A)genes and review the available relevant literature.CASE SUMMARY A 20-year-old man was admitted to our center with a 6-year history of recurrent abdominal pain,diarrhea,and dark stools.At the onset 6 years ago,the patient had received treatment at a local hospital for abdominal pain persisting for 7 d,under the diagnosis of diffuse peritonitis,acute gangrenous appendicitis with perforation,adhesive intestinal obstruction,and pelvic abscess.The surgical treat-ment included exploratory laparotomy,appendectomy,intestinal adhesiolysis,and pelvic abscess removal.The patient’s condition improved and he was dis-charged.However,the recurrent episodes of abdominal pain and passage of black stools started again one year after discharge.On the basis of these features and results of subsequent colonoscopy,the clinical diagnosis was established as in-flammatory bowel disease(IBD).Accordingly,aminosalicylic acid,immunotherapy,and related symptomatic treatment were administered,but the symptoms of the patient did not improve significantly.Further investigations revealed mutations in the ACVRL1 and PLA2G4A genes.ACVRL1 and PLA2G4A are involved in angiogenesis and coagulation,respectively.This suggests that the chronic intestinal ulcers and bleeding in this case may be linked to mutations in the ACVRL1 and PLA2G4A genes.Oral Kangfuxin liquid was administered to promote healing of the intestinal mucosa and effectively manage clinical symptoms.CONCLUSION Mutations in the ACVRL1 and PLA2G4A genes may be one of the causes of chronic intestinal ulcers and bleeding in IBD.Orally administered Kangfuxin liquid may have therapeutic potential.

Hepatitis A virus-associated acute acalculous cholecystitis in an adult-onset Still’s disease patient:A case report and review of the literature

BACKGROUND Acute acalculous cholecystitis(AAC)is inflammation of the gallbladder without evidence of calculi.Although rarely reported,its etiologies include hepatitis virus infection(e.g.,hepatitis A virus,HAV)and adult-onset Still’s disease(AOSD).There are no reports of HAV-associated AAC in an AOSD patient.CASE SUMMARY Here we report a rare case of HAV infection-associated AAC in a 39-year-old woman who had a history of AOSD.The patient presented with an acute abdomen and hypotension.Elevated hepatobiliary enzymes and a thickened and distended gallbladder without gallstones on ultrasonography suggested AAC,but there were no signs of anemia nor thrombocytopenia.Serological screening revealed anti-HAV IgM antibodies.Steroid treatment did not alleviate her symptoms,and she was referred for laparoscopic cholecystectomy.The resected gallbladder was hydropic without perforation,and her clinical signs gradually improved after surgery.CONCLUSION AAC can be caused by HAV in AOSD patients.It is crucial to search for the underlying etiology for AAC,especially uncommon viral causes.

Pulmonary cryptococcosis after immunomodulator treatment in patients with Crohn’s disease:Three case reports

BACKGROUND Corticosteroids and anti-tumor necrosis factorαmAbs are widely used to treat Crohn's disease(CD).However,one disadvantage of this treatment is impairment of normal immune function,leading to an increased risk of infection.Cryptococcus infection is an opportunistic infection that occurs mainly in immunocompromised patients and poses a significant diagnostic challenge in patients with CD.CASE SUMMARY Here,we report three cases of pulmonary cryptococcosis in patients with CD after receiving immunomodulatory treatment.The patients presented with no or mild respiratory symptoms.Chest computed tomography scans revealed pulmonary nodules in the unilateral or bilateral lobes.Diagnoses were made using pathological examination and metagenomic sequencing.The patients were treated with fluconazole 400 mg once daily for 1 to 6 mo,and symptoms were resolved.Literature searches were conducted in PubMed,Web of Science,and Embase to retrieve previously reported cases and summarize patient characteristics.CONCLUSION The incidence of cryptococcus infection has increased along with immunomodulator use.Clinical vigilance is required for early identification and standardized treatment.

Least Privileges and Role’s Inheritance of RBAC

The main advantages of role-based access control （RBAC） are able to support the well-known security principles and roles＇inheritance. But for there remains a lack of specific definition and the necessary formalization for RBAC, it is hard to realize RBAC in practical work. Our contribution here is to formalize the main relations of RBAC and take first step to propose concepts of action closure and deta closure of a role, based on which we got the specification and algorithm for the least privileges of a role. We propose that roles＇ inheritance should consist of inheritance of actions and inheritance of data, and then we got the inheritance of privileges among roles, which can also be supported by existing exploit tools.

Pediatric-type follicular lymphoma in a Crohn’s disease patient receiving anti-α4β7-integrin therapy:A case report

BACKGROUND Patients with autoimmune conditions receiving immunosuppressants are at risk of non-Hodgkin lymphomas(NHL).Vedolizumab(anti-α4β7-integrin antibody),a treatment-of-choice for Crohn’s disease(CD),reduces inflammatory lymphocyte trafficking into the intestinal mucosa.This effect is believed to be confined to the colon.CASE SUMMARY We report the case of a CD patient on vedolizumab for five years who developed pediatric-type follicular lymphoma.Work-up prior to therapy revealed a reduction in circulating T-lymphocytes and their suppressed response to mitogens.Rituximab,cyclophosphamide,vincristine,and prednisone chemoimmunotherapy resulted in durable lymphoma remission,and vedolizumab treatment was continued.While the patient’s T-lymphocyte population and immunoglobulin production recovered,the T-lymphocyte mitogen response remained suppressed.CONCLUSION This patient’s NHL may be linked to receiving anti-α4β7 therapy.Further research could be beneficial to determine if proactive surveillance for NHL and other systemic diseases is indicated in patients on vedolizumab.

Small bowel adenocarcinoma in neoterminal ileum in setting of stricturing Crohn’s disease:A case report and review of literature

BACKGROUND Small bowel adenocarcinomas(SBA) are rare malignancies with exceedingly low survival rates, with different presentation in Crohn’s disease(CD). CD-induced SBA poses diagnostic challenges given overlapping presentation with stricturing CD and lack of diagnostics for early detection. Moreover, guidance is lacking on the impact of recently approved therapeutics in CD on SBA management. Here, we aim to highlight the future of CD-induced SBA management and discuss the potential merit of balloon enteroscopy and genetic testing for earlier detection.CASE SUMMARY We report the case of a 60-year-old female with longstanding Crohn’s ileitis, presenting with acute obstructive symptoms attributed to stricturing phenotype. Her obstructive symptoms were refractory to intravenous(Ⅳ) steroids, with further investigation via computed tomography enterography not providing additional diagnostic yield. Ultimately, surgical resection revealed SBA in the neoterminal ileum, with oncologic therapy plan created. However, this therapy plan could not be initiated due to continued obstructive symptoms attributed to active CD. Ultimately, infused biologic therapy was initiated, but her obstructive symptoms continued to remain dependent on Ⅳ corticosteroids. Review of diagnostics by a multidisciplinary care team suggested metastatic disease in the peritoneum, lending to a shift in the goals of care to comfort.CONCLUSION With the diagnostic and therapeutic challenges of concurrent SBA and CD, multidisciplinary care and algorithmic management can optimize outcomes.

Iatrogenic atlantoaxial rotatory subluxation after thyroidectomy in a pediatric patient:A case report

BACKGROUND Atlantoaxial rotatory subluxation(AARS)is an uncommon disease with a greater prevalence among children than adults,and it is mostly associated with trauma.Iatrogenic spinal injury accounts for a low percentage of injuries.However,in AARS,20%-40%of cases are associated with surgery,and 48%are caused by infection.Here,we describe our experience with a case of iatrogenic AARS after general anesthesia.CASE SUMMARY A 12-year-old girl presented with right-sided torticollis and cervical motion limit.The patient had undergone thyroidectomy 2 mo ago.Computed tomography revealed AARS with bilateral locked facets.Following the failure of repeated external reduction under general anesthesia,the patient underwent an open surgical reduction.The patient gained atlantoaxial alignment without any complications.Follow-up radiographs showed a normal appearance without instability.The cervical spine of children is more predisposed to injury due to anatomical and biomechanical differences.AARS secondary to infection and surgery is known as Grisel’s syndrome,which involves non-traumatic AARS.Several cases of AARS after surgery and other procedures with no evidence of inflammation have been reported.Our experience shows that surgery requiring hyperextension of the neck after general anesthesia should also be included as a risk factor.CONCLUSION Surgeons and anesthesiologists should be careful not to excessively extend the neck during pediatric surgery.Moreover,clinicians caring for pediatric patients with recent head and neck procedures must be aware of common AARS presentations.

Total removal of a large esophageal schwannoma by submucosal tunneling endoscopic resection:A case report and review of literature

BACKGROUND Primary schwannoma is a rare submucosal tumor of the esophagus,which is most often benign,and surgery is the only effective treatment.So far,only a few cases have been reported.Herein,we reported a single case diagnosed with primary esophageal schwannoma that was totally removed by submucosal tunneling endoscopic resection(STER).CASE SUMMARY A 62-year-old man presented to the hospital with a history of resection of a malignant gastric tumor and mild dysphagia.Endoscopic examination revealed a large submucosal elevated lesion in the esophagus 25-30 cm from the incisors.Endoscopic ultrasonography detected a 45 mm×35 mm×31 mm hypoechoic lesion;chest computed tomography showed a mass of approximately 55 mm×35 mm×29 mm.A preliminary examination showed features suggestive of a stromal tumor.Pathological findings indicated esophageal schwannoma.Next,STER alone was performed to completely resect the mass,and the patient recovered well post-surgery.Afterward,the patient was discharged and showed no tumor recurrence at 33 mo of follow-up.CONCLUSION Endoscopic resection is still an effective treatment for large esophageal schwannomas(>30 mm)under meticulous morphological evaluation.

Extraskeletal Ewing sarcoma of the stomach:A rare case report

BACKGROUND Extraskeletal Ewing sarcoma(EES)is a rare and highly malignant small round cell tumor associated with a poor clinical outcome.Ewing sarcoma(ES)involving the stomach is an uncommon presentation and can be easily confused with other small round cell tumors.We herein present a rare case of ES involving the gastric area.CASE SUMMARY We report a case of gastric ES in a 19-year-old female patient who initially presented with a complaint of a tender epigastric mass for 5 d.Contrast-enhanced abdominal computed tomography revealed a soft-tissue-density mass with a diameter of 8.5 cm between the liver and stomach;the mass was connected to the gastric antrum.Then,the mass was surgically excised completely.Upon histopathological,immunophenotype and molecular analysis,the mass was identified to be a primary gastric ES.CONCLUSION EES is an aggressive tumor with poor prognosis.Therefore,early diagnosis and timely intervention are essential for a good prognosis.It is imperative for us to raise awareness about this rare tumor.Surgical resection is still the best treatment option.

Recovery from Bell’s palsy after treatment using uncultured umbilical cord-derived mesenchymal stem cells:A case report

BACKGROUND Bell’s palsy is an idiopathic facial palsy with an unknown cause,and 75%of patients heal spontaneously.However,the other 25%of patients continue experiencing mild or severe disabilities,resulting in a reduced quality of life.Currently,various treatment methods have been developed to treat this disease.However,there is controversy regarding their effectiveness,and new alternative treatments are needed.CASE SUMMARY The patient suffered from left-sided facial paralysis due to Bell’s palsy for 7 years.The patient received an uncultured umbilical cord-derived mesenchymal stem cell transplant eight times for treatment.After follow-up for 32 mo,the paralysis was cured,and there was no recurrence.CONCLUSION Uncultured umbilical cord-derived mesenchymal stem cell transplantation may be a potential treatment for patients with Bell’s palsy who do not spontaneously recover.

Adult localized Langerhans cell histiocytosis:A case report

BACKGROUND Langerhans cell histiocytosis(LCH)is a rare clonal proliferative disease of Langerhans cells with unknown pathogenesis.An increasing number of clinicians recognize that LCH has a wide clinical spectrum and a highly varied course.Adults rarely develop LCH.Here,we report a case of adult localized LCH.CASE SUMMARY A 32-year-old woman presented with plaques and ulcers on the vulva and crissum,accompanied by pain that persisted for more than one year.Physical examination revealed a red-infiltrating plaque with ulcerations and exudates in the vulva and crissum.Pathological examination revealed a diffuse infiltration of lymphocytes,eosinophilic granulocytes,and histiocytoid cells in the superficial dermis.Proliferative histiocytoid cells showed mild atypia,partly with kidneyshaped nuclei.Immunohistochemical examination showed that the histiocytoid cells were positive for S100 protein and CD1 and weakly positive for CD68(20%+),with a Ki-67 index of 30%.Laboratory tests did not reveal any other systemic damage.The patient was diagnosed with adult localized LCH and was prescribed oral prednisone(20 mg)once daily.The skin lesions gradually improved and are still being followed-up.CONCLUSION Adult localized LCH is rare and must be differentiated from other common conditions.

Atypical case of bow hunter’s syndrome linked to aberrantly coursing vertebral artery: A case report

BACKGROUND In bow hunter’s syndrome(BHS),also known as rotational vertebral artery(VA)syndrome,there is dynamic/rotational compression of the VA producing verte-brobasilar insufficiency.Most occurrences involve atlantoaxial rather than mid-cervical VA compromise,the latter being rarely reported.Herein,we detail successful VA decompression at mid-cervical spine,given a departure from its usual course.CASE SUMMARY The patient,a 45-year-old man,presented to our hospital with occipital headache and vertigo.Computed tomography angiography showed anomalous C4 entry of right VA,with compression upon head rotation to that side.Thyroid cartilage and anterior tubercle of C5 transverse process were visibly at fault.We opted for sur-gery,using an anterior cervical approach to remove the anterior tubercle.Patient recovery was uneventful and brought resolution of all preoperative symptoms.CONCLUSION BHS is an important consideration where aberrant coursing of VA and neurologic symptoms coexist.

Autoimmune hepatitis and eosinophilia:A rare case report

BACKGROUND Autoimmune hepatitis consists of a chronic liver disease whose etiology is unknown.It is comprised of relevant immunological aspects and of immunemediated liver injury.Eosinophilia may be a considerable feature,particularly happening in male patients.CASE SUMMARY We report here a Crohn´s disease patient presenting with de novo hypergammaglobulinemia,circulating autoantibodies and elevated transaminase levels.He also had significant peripheral eosinophilia and elevated immunoglobulin E levels at diagnosis.The pathology findings from liver biopsy were compatible with autoimmune hepatitis with eosinophilic infiltration.CONCLUSION This is the first report of autoimmune hepatitis with exuberant eosinophilic infiltration in the liver and bone marrow,described in a patient with Crohn’s disease.

Huge Bartholin’s cyst managed by primary marsupialization:A case report

BACKGROUND We report on the case of a 21-year-old female who could not walk due to a huge Bartholin's cyst occupying the right vulvar region.The patient was treated by marsupialization of the Bartholin’s cyst and postoperative antibiotic therapy.CASE SUMMARY A 21-year-old female without a medical history of sexually transmitted diseases presented to our outpatient clinic with complaints of a painful vulvar lump and a green-yellowish discharge for 4 d.A large Bartholin’s cyst,10 cm in diameter,was suspected over the right side of the vulva.She was advised to be admitted to the ward for intravenous antibiotic treatment and to perform marsupialization several days later.However,the patient requested immediate surgery because of time limitations.The patient underwent marsupialization under local anesthesia.The procedure was well-tolerated.After making the incision,a significant amount of malodorous pus(approximately 30 mL)was removed from the abscess.The patient was followed up for 1 wk postoperatively.The Bartholin’s cyst regressed,and the surgical wound healed well.CONCLUSION Our case shows that an initial marsupialization followed by oral antibiotic therapy can be an appropriate treatment option for a large Bartholin’s cyst.However,depending on the patient’s condition,a suitable treatment,either cystectomy or marsupialization,can be considered.