Unusual ovarian hyperstimulation syndrome presentation:Pleural effusion without ascites.A case report

BACKGROUND Ovarian hyperstimulation syndrome(OHSS)is a life-threatening complication that can occur in the luteal phase or early pregnancy after controlled ovarian stimulation.This case report highlights a unique manifestation of OHSS involving pleural effusion(PE)in a patient without identifiable risk factors.CASE SUMMARY A 39-year-old woman who underwent controlled ovarian hyperstimulation for an in vitro fertilization(IVF)cycle experienced dyspnea on the eleventh day of post oocyte retrieval.The diagnosis was severe OHSS with a unique manifestation of PE without ascites.Clinical management involved fluid balance and treatment with albumin,furosemide,thromboembolic prophylaxis,and thoracentesis.A continued drainage of the pleural cavity was performed.The patient had a favo-rable outcome,and a dichorionic diamniotic gestation passed without incident.CONCLUSION OHSS and its potential complications can include respiratory distress and PE,as well as thromboembolic disorders.

Saccharomyces boulardii as a single trigger of food protein-induced enterocolitis syndrome: Seven case reports

BACKGROUND Food protein-induced enterocolitis syndrome(FPIES)is the most serious type of non-immunoglobulin E(IgE)-mediated food allergic reaction manifesting as sepsis-like symptom,which can lead to shock.Saccharomyces boulardii(S.boulardii),a probiotic prescribed frequently in clinical settings,has been reported to trigger FPIES in an infant with soy-triggered FPIES.In this report,we describe a new clinical FPIES in which S.boulardii was the sole triggering factor of acute FPIES adverse reaction in seven healthy infants.CASE SUMMARY Seven FPIES cases triggered by only S.boulardii were gathered from 2011 to the present.None of the patients had previously experienced any allergic reaction to cow’s milk,soy,or complementary food.The age of the patients was 4-10-months old,and the symptoms of FPIES developed after ingestion of S.boulardii,which is mostly prescribed for the treatment of gastroenteritis or antibiotic-associated diarrhea.All patients experienced severe repetitive vomiting 1-3 hours after S.boulardii ingestion.Extreme lethargy,marked pallor,and cyanosis were also observed.No IgE-mediated hypersensitivity developed in any patient.Diarrhea was followed by initial intense vomiting in approximately 5-10 hours after S.boulardii ingestion,and only one case showed bloody,purulent,and foul-smelling diarrhea.The patients stabilized quickly,mostly within 6 hours.Symptoms got all improved within 24 hours after discontinuation of S.boulardii.CONCLUSION S.boulardii can be the sole trigger of acute FPIES and be prescribed cautiously even in healthy children without FPIES.

Anlotinib-induced sick sinus syndrome:Two case reports

BACKGROUND This manuscript describes the first known cases of sick sinus syndrome(SSS)associated with the use of anlotinib in non-small cell lung cancer patients,highlighting the need for increased vigilance and cardiac monitoring.CASE SUMMARY Two patients with non-small cell lung cancer developed SSS after 15 months and 5 months of anlotinib treatment,respectively,presenting with syncope and palpit-ations.Electrocardiogram confirmed SSS,and different treatment approaches were taken for each patient.One patient received a dual-chamber permanent pacemaker,while the other discontinued the medication and experienced symptom resolution.CONCLUSION Anlotinib can induce SSS,suggesting that cardiac monitoring is crucial during anlotinib treatment.Individualized management strategies are necessary for affected individuals.

Cauda equina syndrome with urinary retention as a postoperative complication of lumbar spine surgery:A case report

BACKGROUND Cauda equina syndrome(CES)is characterized by a group of symptoms that may be caused by inflammation,spinal cord compression,venous congestion,or ischemia.This syndrome is commonly an indication for surgical intervention but has not been determined as a postoperative complication following surgery for lumbar spine disease.CASE SUMMARY To report the case of a 54-year-old male patient who had CES following spinal surgery,with no obvious compression lesions found during re-exploration,suggesting that vascular insufficiency may have contributed to the condition.Furthermore,a series of urodynamic studies on bladder recovery patterns in such complications have also been investigated.CONCLUSION Postoperative CES requires urgent imaging and exploration to rule out compression;noncompressive cases,including vascular insufficiency may performed conservative management.

Pembrolizumab-induced Guillain-Barrésyndrome in triple-negative breast cancer:A case report

BACKGROUND The programmed cell death protein 1 inhibitor pembrolizumab has become a key treatment for various cancers,including triple-negative breast cancer.However,it is associated with immune-related adverse events,including rare but serious neurological complications such as Guillain-Barrésyndrome(GBS).GBS is a potentially life-threatening autoimmune disorder characterized by muscle weakness and paralysis.We present a unique case of pembrolizumab-induced GBS to highlight the importance of recognizing this complication and managing it promptly in patients receiving immune checkpoint inhibitors.CASE SUMMARY A 69-year-old woman with a medical history of hypertension,anxiety,depression,and stage IIIB triple-negative breast cancer treated with pembrolizumab,carboplatin,and paclitaxel,presented to the emergency department with a 1-month history of tingling,lower extremity weakness,and shooting pain.Symptoms progressed to global weakness,ascending paralysis,and double vision.Neurological examination revealed significant lower extremity weakness and sensory deficits.Magnetic resonance imaging of the lumbar spine and cerebrospinal fluid analysis confirmed GBS.Initial treatment with intravenous immunoglobulin led to relapse,requiring additional intravenous immunoglobulin and high-dose glucocorticoids.The patient’s condition improved,pembrolizumab therapy was permanently discontinued,and she was discharged to a rehabilitation facility.CONCLUSION Pembrolizumab can induce GBS,necessitating early recognition,prompt diagnosis,and multidisciplinary management to prevent serious complications.

Tricuspid mass-curious case of Li-Fraumeni syndrome: A case report

BACKGROUND Li-Fraumeni syndrome(LFS)is a rare autosomal dominant cancer-predisposing syndrome,which can manifest as a polymorphic spectrum of malignancies.LFS is associated with an early onset in life,with the majority of cases occurring prior to the age of 46.Notwithstanding the infrequency of primary cardiac tumors,it behooves clinicians to remain vigilant in considering the differential diagnosis of such tumors in LFS patients who present with a cardiac mass.This is due to the markedly elevated risk for malignancy in this particular population,far surpassing that of the general populace.CASE SUMMARY Herein,we present a case of a 30-year-old female with LFS who was found to have a tricuspid valve leaflet mass.CONCLUSION This case exemplifies valuable learning points in the diagnostic approach for this exceptionally rare patient population.

Pulmonary artery stent thrombosis and symptomatic pulmonary hypertension following COVID-19 infection in Alagille patient:A case report

BACKGROUND Alagille syndrome is a multisystem disease that results in various vascular anomalies,commonly involving the cardiac and pulmonary systems.To the best of our knowledge,there is no literature regarding the cardiovascular outcomes of these patients in association with coronavirus disease 2019(COVID-19).CASE SUMMARY A 34-year-old woman with a history of Alagille syndrome who underwent successful atrial septal defect with partial anomalous pulmonary veins and patent ductus arteriosus repair,as well as left pulmonary artery catheterization and stenting in childhood due to pulmonary stenosis.The patient was without any respiratory symptoms and was a dancer prior to contracting COVID-19.Several weeks after her COVID-19 infection,she developed left pulmonary artery stent thrombosis and subsequent symptomatic pulmonary hypertension.A treatment strategy of anticoagulation alongside pharmacological agents for pulmonary hypertension for 3 months followed by balloon pulmonary artery angioplasty to reopen the stenosis was unsuccessful.CONCLUSION In the era of COVID-19,patients with pulmonary vascular malformations and endovascular stents are at an increased risk for chronic thromboembolic disease.Patients may benefit from prophylactic antiplatelet or anticoagulation therapy.Stent thrombosis is a devastating phenomenon and should be treated urgently and aggressively with balloon pulmonary angioplasty,and/or a thrombolytic agent.

Cytokine release syndrome triggered by programmed death 1 blockade(sintilimab)therapy in a psoriasis patient:A case report

BACKGROUND In recent years,immune checkpoint inhibitors(ICIs)have demonstrated remarkable efficacy across diverse malignancies.Notably,in patients with advanced gastric cancer,the use of programmed death 1(PD-1)blockade has significantly prolonged overall survival,marking a pivotal advancement comparable to the impact of Herceptin over the past two decades.While the therapeutic benefits of ICIs are evident,the increasing use of immunotherapy has led to an increase in immune-related adverse events.CASE SUMMARY This article presents the case of a patient with advanced gastric cancer and chronic plaque psoriasis.Following sintilimab therapy,the patient developed severe rashes accompanied by cytokine release syndrome(CRS).Fortunately,effective management was achieved through the administration of glucocorticoid,tocilizumab,and acitretin,which resulted in favorable outcomes.CONCLUSION Glucocorticoid and tocilizumab therapy was effective in managing CRS after PD-1 blockade therapy for gastric cancer in a patient with chronic plaque psoriasis.

Uveitis glaucoma hyphema syndrome as a result of glaucoma implant: A case report

BACKGROUND Glaucoma is caused by increased intraocular pressure(IOP)that damages the optic nerve,leading to blindness.The Ahmed glaucoma valve(AGV)is a glau-coma drainage implant device that is used in glaucoma patients with uncontrolled IOP.A possible complication after any ocular surgery however is hyphema,which can itself progress to uveitis glaucoma hyphema(UGH)syndrome on rare occasions.UGH syndrome has not yet been reported as a complication of AGV implantation.CASE SUMMARY Here,we have reported a case of a 55-year-old female who developed both hyphema and pigmentation as a result of AGV implantation.We confirmed UGH syndrome secondary to AGV implantation after the patient underwent another surgery to shorten and reposition the AGV tube.After the second surgery,the patient’s IOP was reduced,and she had a clear cornea and no signs of hyphema.CONCLUSION This first report of UGH syndrome as a complication of AGV implantation reminds clinicians that frequent follow-up is paramount.

Abdominal cocoon syndrome-a rare culprit behind small bowel ischemia and obstruction:Three case reports

BACKGROUND Abdominal cocoon syndrome(ACS)represents a category within sclerosing encapsulating peritonitis,characterized by the encapsulation of internal organs with a fibrous,cocoon-like membrane of unknown origin,resulting in bowel obstruction and ischemia.Diagnosing this condition before surgery poses a cha-llenge,often requiring confirmation during laparotomy.In this context,we depict three instances of ACS:One linked to intestinal obstruction,the second exclu-sively manifesting as intestinal ischemia without any obstruction,and the final case involving a discrepancy between the radiologist and the surgeon.CASE SUMMARY Three male patients,aged 53,58,and 61 originating from Northern Thailand,arrived at our medical facility complaining of abdominal pain without any prior surgeries.Their vital signs remained stable during the assessment.The diagnosis of abdominal cocoon was confirmed through abdominal computed tomography(CT)before surgery.In the first case,the CT scan revealed capsules around the small bowel loops,showing no enhancement,along with mesenteric congestion affecting both small and large bowel loops,without a clear obstruction.The second case showed intestinal obstruction due to an encapsulated capsule on the CT scan.In the final case,a patient presented with recurring abdominal pain.Initially,the radiologist suspected enteritis as the cause after the CT scan.However,a detailed review led the surgeon to suspect encapsulating peritoneal sclerosis(ACS)and subsequently perform surgery.The surgical procedure involved complete removal of the encapsulating structure,resection of a portion of the small bowel,and end-to-end anastomosis.No complications occurred during surgery,and the patients had a smooth recovery after surgery,eventually discharged in good health.The histopathological examination of the fibrous membrane(cocoon)across all cases consistently revealed the presence of fibro-collagenous tissue,without any indications of malignancy.CONCLUSION Individuals diagnosed with abdominal cocoons commonly manifest vague symptoms of abdominal discomfort.An elevated degree of clinical suspicion,combined with the application of appropriate radiological evaluations,markedly improves the probability of identifying the abdominal cocoon before surgical intervention.In cases of complete bowel obstruction or ischemia,the established norm is the comprehensive removal of the peritoneal sac as part of standard care.Resection with intestinal anastomosis is advised solely when ischemia and gangrene have been confirmed.

Clinical and genetic characteristics of a child with Sotos syndrome and attention-deficit/hyperactivity disorder:A case report

BACKGROUND Sotos syndrome is an autosomal dominant disorder,whereas attention-deficit/hyperactivity disorder(ADHD)is a neurodevelopmental condition.This report aimed to summarize the clinical and genetic features of a pediatric case of Soros syndrome and ADHD in a child exhibiting precocious puberty.CASE SUMMARY The patient presented with accelerated growth and advanced skeletal maturation;however,she lacked any distinct facial characteristics related to specific genetic disorders.Genetic analyses revealed a paternally inherited heterozygous synonymous mutation[c.4605C>T(p.Arg1535Arg)].Functional analyses suggested that this mutation may disrupt splicing,and bioinformatics analyses predicted that this mutation was likely pathogenic.After an initial diagnosis of Sotos syndrome,the patient was diagnosed with ADHD during the follow-up period at the age of 8 years and 7 months.CONCLUSION The potential for comorbid ADHD in Sotos syndrome patients should be considered to avoid the risk of a missed diagnosis.

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.

Cronkhite-Canada syndrome with esophagus involvement and sixyear follow-up:A case report

BACKGROUND Cronkhite-Canada syndrome(CCS)is a rare,noninherited disease characterized by gastrointestinal polyposis with diarrhea and ectodermal abnormalities.CCS polyps are distributed through the whole digestive tract,and they are common in the stomach and colon but very uncommon in the esophagus.CASE SUMMARY Here,we present a case of a 63-year-old man with skin hyperpigmentation accompanied by diarrhea,alopecia,and loss of his fingernails.Laboratory data indicated anemia,hypoalbuminemia,hypocalcemia,hypokalemia,and positive fecal occult blood.Endoscopy showed numerous polyps scattered throughout the digestive tract,including the esophagus.He was treated with nutritional support and glucocorticoids with remission of his symptoms.CONCLUSION Comprehensive treatment led by hormonal therapy can result in partial or full remission of clinical symptoms.Treatment should be individualized for each patient according to their therapy response.Surveillance endoscopy is necessary for assessing mucosal disease activity and detecting malignant transformation.

Clinical evolution of antisynthetase syndrome-associated interstitial lung disease after COVID-19 in a man with Klinefelter syndrome:A case report

BACKGROUND This study presents a case of rapidly developing respiratory failure due to antisynthetase syndrome(AS)following coronavirus disease 2019(COVID-19)in a 33-year-old man diagnosed with Klinefelter syndrome(KS).CASE SUMMARY A 33-year-old man with a diagnosis of KS was admitted to the Department of Pulmonary and Critical Care Medicine of a tertiary hospital in China for fever and shortness of breath 2 wk after the onset of COVID-19.Computed tomography of both lungs revealed diffuse multiple patchy heightened shadows in both lungs,accompanied by signs of partial bronchial inflation.Metagenomic next-generation sequencing of the bronchoalveolar lavage fluid suggested absence of pathogen.A biopsy specimen revealed organizing pneumonia with alveolar septal thickening.Additionally,extensive auto-antibody tests showed strong positivity for anti-SSA,anti-SSB,anti-Jo-1,and anti-Ro-52.Following multidisciplinary discussions,the patient received a final diagnosis of AS,leading to rapidly progressing respiratory failure.CONCLUSION This study underscores the clinical progression of AS-associated interstitial lung disease subsequent to viral infections such as COVID-19 in patients diagnosed with KS.

HDR syndrome presented with nephrotic syndrome in a Chinese boy: A case report

BACKGROUND HDR syndrome is a rare genetic disease caused by variants in the GATA3 gene and is phenotypically defined by the triad of hypoparathyroidism(H),deafness(D),and renal disease(R).Renal disorders of HDR are mainly developmental ab-normalities,although renal functional abnormalities can also be observed.Ne-phrotic syndrome or nephrotic-level proteinuria is rare in HDR syndrome.Here,we report a Chinese infant with HDR syndrome who presented with early-onset nephrotic syndrome.We suggest that variants in the GATA3 gene might be asso-ciated with nephrotic syndrome.(p.Pro235 Leu),in exon 3 of the GATA3 gene.CONCLUSION We report an infant with HDR syndrome who presented with early-onset nephrotic syndrome in China.We suggest that variants in the GATA3 gene might be associated with infant-onset nephrotic syndrome.

Polyneuropathy, organomegaly, endocrinopathy, M-protein, skin changes syndrome with dilated cardiomyopathy: A case report

BACKGROUND Polyneuropathy,organomegaly,endocrinopathy,M-protein,skin changes(POEMS)syndrome is a rare paraneoplastic syndrome that encompass multiple systems.The most common clinical symptoms of POEMS syndrome are pro-gressive sensorimotor polyneuropathy,organ enlargement,endocrine disorders,darkening skin,a monoclonal plasma cell proliferative disorder,and lymph node hyperplasia.The organomegaly consists of hepatosplenomegaly and/or lym-phadenopathy;cases of cardiomyopathy are rare.Diagnoses are often delayed because of the atypical nature of the syndrome,exposing patients to possibly severe disability.Therefore,identifying atypical symptoms can improve the prognosis and quality of life among POEMS syndrome patients.lenalidomide and dexamethasone.CONCLUSION When patients with cardiomyopathy have systemic manifestations such as numb limbs and darkening skin,the POEMS syndrome is the most possible diagnosis.

Moyamoya syndrome may result from psoriasis: Four case reports

BACKGROUND Moyamoya syndrome(MMS)is a group of diseases that involves more than one underlying disease and is accompanied by moyamoya vascular phenomena.Psoriasis is a chronic immune skin disease closely linked to high blood pressure and heart disease.However,psoriasis-related MMS has not been reported.CASE SUMMARY We collected data on patients with stroke due to MMS between January 2017 and December 2019 and identified four cases of psoriasis.Case histories,imaging,and hematological data were collected.The average age of the initial stroke onset was 58.25±11.52 years;three cases of hemorrhagic and one case of ischemic stroke were included.The average duration from psoriasis confirmation to the initial MMS-mediated stroke onset was 17±3.56 years.All MMS-related stenoses involved the bilateral cerebral arteries:Suzuki grade III in one case,grade IV in two cases,and grade V in one case.Abnormally elevated plasma interleukin-6 levels were observed in four patients.Two patients had abnormally elevated immunoglobulin E levels,and two had thrombocytosis.All four patients received medication instead of surgery.With an average follow-up time of 2 years,two causing transient ischemic attacks occurred in two patients,and no hemorrhagic events occurred.CONCLUSION Psoriasis may be a potential risk factor for MMS.Patients with psoriasis should be screened for MMS when they present with neurological symptoms.

Sustained remission of Cronkhite-Canada syndrome after corticosteroid and mesalazine treatment: A case report

BACKGROUND Cronkhite-Canada syndrome(CCS)is a rare disease of unknown etiology.The optimal treatment for CCS remains unknown.Treatment with corticosteroids is considered the mainstay treatment because of its high efficacy,but the therapeutic strategy for steroid-resistant CCS is not yet established.CASE SUMMARY This is the case of an 81-year-old woman who was diagnosed with CCS.Given her severe diarrhea,nausea,vomiting,and hypoproteinemia,hormone therapy(40 mg/d)was administered,and the symptoms improved within 1 wk.After 3 mo,the patient had no obvious symptoms.The polyps were significantly reduced on review gastroscopy and colonoscopy,thus hormone reduction gradually began.The hormone level was maintained at 10 mg/d after 6 mo.Despite the age of the patient and the side effects of hormones,the patient had no obvious discomfort.However,hormone drugs were discontinued,and mesalazine was administered orally at 3 g/d.The patient's symptoms continued to improve after a follow-up of 5 years.CONCLUSION Corticosteroids and mesalazine are potential treatment options for CCS.

Antiphospholipid syndrome presenting as recurrent coronary thrombosis:A case report

BACKGROUND Antiphospholipid syndrome(APS)is a chronic autoimmune disease characterized by venous or arterial thrombosis,pregnancy morbidity and a variety of other autoimmune and inflammatory complications.Here,we report a case of APS associated with multiple coronary thromboses.CASE SUMMARY The patient,a 28-year-old male,suffered from recurrent coronary thromboses over a period of 31 months.Despite undergoing interventional coronary procedures,thrombolytic therapy,and anticoagulation treatment,the condition persisted intermittently.An extensive search for underlying thrombogenic factors revealed a diagnosis of APS.Accurate adjustment of the medication regimen led to the absence of further acute coronary syndrome(ACS)episodes during the subsequent 20-month follow-up.Although the patient occasionally experiences chest tightness,no further symptoms of distress have been reported.CONCLUSION APS can manifest as ACS.Screening for rheumatologic and immunological conditions is essential when encountering patients with multiple coronary thromboses.Treatment strategy should include symptomatic relief and a targeted and aggressive approach to address the underlying pathophysiology.

Subsequent bilateral acute carpal tunnel syndrome due to tophaceous infiltration:A case report

BACKGROUND Acute carpal tunnel syndrome(ACTS)is commonly caused by repetitive strain,trauma,or inflammatory conditions.However,ACTS due to tophaceous gout is a clinical event that remains poorly understood and underreported.This rare manifestation necessitates prompt diagnosis and intervention to prevent irreversible complications.CASE SUMMARY A 51-year-old man who had poorly controlled hyperuricemia presented with ACTS secondary to tophaceous gout.Because of rapid symptom progression symptoms and severe median nerve compression within 3 mo,the patient underwent emergency decompression surgery for both wrists at different time points.Postoperatively,he exhibited complete recovery of sensory and motor functions,with no recurrence at long-term follow-up.Favorable outcomes were achieved through immediate decompression surgery,anti-inflammatory medications,postoperative active and passive range-of-motion exercises,and intermittent wrist splinting.Prompt diagnosis and surgical intervention,when necessary,are crucial for preventing long-term complications and obtaining favorable outcomes in patients with ACTS.An optimal gout management strategy involving pharmacologic therapy and lifestyle modifications may help minimize ACTS recurrence and improve clinical outcomes.CONCLUSION Prompt surgical intervention and optimal gout management are crucial for preventing irreversible nerve damage and ACTS recurrence.