BACKGROUND The incidence of malignant gastrointestinal(GI)tumors is increasing,and advancements in medical care have significantly improved patient survival rates.As a result,the number of cases involving multiple pri...BACKGROUND The incidence of malignant gastrointestinal(GI)tumors is increasing,and advancements in medical care have significantly improved patient survival rates.As a result,the number of cases involving multiple primary cancers(MPC)has also increased.The rarity of MPC and the absence of sensitive and specific dia-gnostic markers often lead to missed or incorrect diagnoses.It is,therefore,of vital importance to improve the vigilance of clinicians and the accurate diagnosis of this disease.Patients with GI malignancies face a higher relative risk of deve-loping additional primary malignant tumors compared to those with other systemic tumors.Vigilant monitoring and follow-up are crucial,especially for high-risk groups,which include older adults,men,those with addictions to alcohol and tobacco,those with a family history of tumors,and those who have undergone radiotherapy.CASE SUMMARY In this article,we report three cases of MPC,each involving malignant tumors of the GI tract as the initial primary carcinoma,offering insights that may aid in effectively managing similar cases.CONCLUSION Patients with GI malignancies face a higher MPC risk.Developing screening and follow-up protocols may enhance detection and treatment outcomes.展开更多
BACKGROUND The liver is the most common site of digestive system tumor metastasis,but not all liver metastases can be traced back to the primary lesions.Although it is unusual,syphilis can impact the liver,manifesting...BACKGROUND The liver is the most common site of digestive system tumor metastasis,but not all liver metastases can be traced back to the primary lesions.Although it is unusual,syphilis can impact the liver,manifesting as syphilitic hepatitis with inflammatory nodules,which might be misdiagnosed as metastasis.CASE SUMMARY This case report involves a 46-year-old female who developed right upper abdominal pain and intermittent low fever that persisted for more than three months.No definitive diagnosis of a tumor had been made in the past decades,but signs of multiple liver metastases were recognized after a computed tomo-graphy scan without evidence of primary lesions.With positive serological tests for syphilis and a biopsy of the liver nodules,a diagnosis of hepatic syphilis was made and confirmed with follow-up nodule reduction after anti-syphilis therapy.CONCLUSION Clinicians must be aware of the possibility that syphilis can cause hepatic inflam-matory masses,especially when liver metastasis is suspected without evidence of primary lesions.A definitive diagnosis should be established in conjunction with a review of the patient’s medical history for accurate therapeutic intervention.展开更多
BACKGROUND Jejunal diverticula(JD)are rare clinical conditions that are typically incidentally detected and asymptomatic.When acute complications arise,surgical exploration may be necessary for accurate diagnosis and ...BACKGROUND Jejunal diverticula(JD)are rare clinical conditions that are typically incidentally detected and asymptomatic.When acute complications arise,surgical exploration may be necessary for accurate diagnosis and appropriate treatment.In this report,we present a case of multiple JD complicated by gastrointestinal bleeding and review the pathogenesis,diagnosis,and treatment of JD to increase clinician awareness of this condition.CASE SUMMARY A 70-year-old male patient with multiple JD presented with repeated massive gastrointestinal bleeding.The patient did not respond to symptomatic conservative treatment.Additional diagnostic investigations,including digestive endoscopy and abdominal angiography,did not reveal any relevant abnormalities.An exploratory laparotomy was subsequently performed,during which a segment of the bowel containing numerous diverticulum-like structures was surgically removed.Following successful discharge from the hospital,the patient did not experience any further episodes of gastrointestinal bleeding during subsequent follow-up.CONCLUSION Complications caused by JD are often difficult to diagnose,and surgical exploration is sometimes the most appropriate method.展开更多
BACKGROUND Ganglioneuroma is a rare,well-differentiated,slow-growing benign tumor of the peripheral nerves,with surgical resection being the only curative treatment.Surgical resection of ganglioneuromas encasing major...BACKGROUND Ganglioneuroma is a rare,well-differentiated,slow-growing benign tumor of the peripheral nerves,with surgical resection being the only curative treatment.Surgical resection of ganglioneuromas encasing major blood vessels remains a substantial clinical challenge.Traditionally,these cases often require open abdominal surgery or combined organ resections,and in some instances,the tumors are considered unresectable.Currently,no reports have described the resection of such tumors via laparoscopy.CASE SUMMARY A 35-year-old woman was admitted to our hospital after the incidental discovery of a retroperitoneal space-occupying lesion.Imaging revealed a mass with the celiac axis and superior mesenteric artery passing through it.A neurogenic tumor was suspected,with ganglioneuroma being the most likely diagnosis.Following comprehensive preoperative preparation,the retroperitoneal tumor was resected using a three-dimensional laparoscopy combined with an organ suspension technique.The surgical approach involved incising the tumor along the vascular axis and conducting meticulous,vascular-preserving tumor excision.The operation lasted approximately 458 minutes,with an estimated blood loss of 50 mL.The patient was discharged on the 8th postoperative day.A transient liver injury occurred after surgery but improved rapidly.After 11 months of postoperative follow-up,no complications or tumor recurrence were observed.CONCLUSION This case illustrates the feasibility of minimally invasive laparoscopic resection for retroperitoneal ganglioneuromas encasing major blood vessels.展开更多
BACKGROUND Gastric bezoars are indigestible masses that can lead to gastrointestinal ob-struction and ulceration.Standard treatments include endoscopic mechanical lithotripsy with a polypectomy snare and Coca-Cola dis...BACKGROUND Gastric bezoars are indigestible masses that can lead to gastrointestinal ob-struction and ulceration.Standard treatments include endoscopic mechanical lithotripsy with a polypectomy snare and Coca-Cola dissolution therapy or a combination of both approaches.However,giant bezoars frequently require multiple treatment sessions and extended hospital stays.Additionally,snare-based mechanical fragmentation may be limited by factors such as bezoar size,shape,density,slipperiness,and restricted working space.In cases where refra-ctory giant bezoars are unresponsive to traditional methods,surgical intervention is often necessary.CASE SUMMARY A 57-year-old male with a history of type 2 diabetes presented with severe epigastric pain and vomiting.Endoscopy revealed two large phytobezoars and a gastric ulcer.Initial attempts at mechanical fragmentation with a polypectomy snare and Coca-Cola ingestion for dissolution were unsuccessful due to the large size and complex structure of the bezoars.An innovative approach using snare-tip electrocautery was then employed.It successfully penetrated the slippery,hard surface of the bezoars and fragmented them into smaller pieces.The patient was subsequently treated with Coca-Cola ingestion,enzyme supplements,and proton pump inhibitors.He was discharged without complications following the endoscopic sessions.CONCLUSION Snare-tip electrocautery is a safe,cost-effective,and minimally invasive alter-native for managing large,refractory gastric bezoars.This is a valuable option in resource-limited settings.展开更多
BACKGROUND Gastric neuroendocrine carcinomas(NECs)exhibit aggressive features,such as rapid growth,higher rate of metastasis,and a generally unfavorable prognosis compared to gastric adenocarcinoma.As a result,therape...BACKGROUND Gastric neuroendocrine carcinomas(NECs)exhibit aggressive features,such as rapid growth,higher rate of metastasis,and a generally unfavorable prognosis compared to gastric adenocarcinoma.As a result,therapeutic options for NECs remain limited,contributing to the poor prognosis of patients.Immunotherapy has emerged as a promising treatment strategy and demonstrated the potential to partially improve the survival and prognosis of patients with NECs.Nevertheless,the unique clinical response termed pseudoprogression(PsP)has garnered considerable attention in the context of immunotherapy.CASE SUMMARY Presented here is a case of NEC recurrence five and a half months after radical gastric surgery.The 45-year-old male patient underwent combination treatment involving a PD-1 blocker and tyrosine kinase inhibitors and encountered two instances of PsP during treatment.The patient ultimately achieved a durable treatment response without altering his treatment regimens,resulting in a substantial therapeutic benefit.CONCLUSION This case report aimed to provide the authors’experience with the diagnosis of PsP and treatment strategies for PsP in ongoing immunotherapy.展开更多
BACKGROUND Here,we report a case of jejunal diverticulosis from an anatomical and histological view.During the“Gross Anatomy course,”we found multiple jejunal diverticula along a total length of 208 cm of intestine....BACKGROUND Here,we report a case of jejunal diverticulosis from an anatomical and histological view.During the“Gross Anatomy course,”we found multiple jejunal diverticula along a total length of 208 cm of intestine.CASE SUMMARY After opening the intestinal tract,we counted 232 jejunal diverticulum entry points with a diameter of up to 2 cm and observed connections between the diverticula that created shortcuts between two distinct intestinal parts.Interestingly,we observed an extreme longitudinal striation on the intestinal parts hosting diverticula.Thorough vessel preparation utilizing a dissecting microscope confirmed that all investigated arteriae rectae ended in a diverticulum.Histological and immunohistochemical investigations revealed that intestinal villi of diverticula were smaller and less prominent than control tissue and that the stratum longitudinale,as well as the stratum circular,were much thinner in the diverticula compared to control tissue.Neither submucosal nor mesenteric plexus could be detected in the diverticula.However,vasoactive intestinal peptidepositive nerve fibers and villin-positive brush border could only be detected in control tissue.This indicates that jejunal diverticulosis is associated with abnormalities of the smooth muscles and a disorder of innervation.CONCLUSION Jejunal diverticulosis originates from mesenteric vessels,featuring smooth muscle changes,absent innervation,and thinning of tissue layers.展开更多
BACKGROUND Systemic lupus erythematosus(SLE)can affect multiple organs or systems.The involvement of the central nervous system can result in the manifestation of epilepsy,an acute confusional state,and other rare neu...BACKGROUND Systemic lupus erythematosus(SLE)can affect multiple organs or systems.The involvement of the central nervous system can result in the manifestation of epilepsy,an acute confusional state,and other rare neuropsychiatric presentations,such as catatonia.CASE SUMMARY We present a case of an adolescent male patient with first-onset SLE who presented with neuropsychiatric symptoms including epilepsy and delirium.The initial utilization of olanzapine to alleviate symptoms of agitation precipitated the emergence of catatonia,which was mitigated by discontinuing olanzapine and supplementing with lorazepam.In this case,whether the catatonia was secondary to the utilization of antipsychotics or to an organic disease is a question that warrants differential diagnosis.CONCLUSION Multidisciplinary collaborative management is the cornerstone for the successful management of severe cases of SLE.展开更多
BACKGROUND Multiple primary malignant tumors refer to the occurrence of two or more primary malignant tumors in the same organ or multiple organs or tissues at the same time or successively in the same patient,and can...BACKGROUND Multiple primary malignant tumors refer to the occurrence of two or more primary malignant tumors in the same organ or multiple organs or tissues at the same time or successively in the same patient,and can occur anywhere in the body.The treatment guidelines for patients with multiple primary malignant tumors are currently controversial.CASE SUMMARY A 51-year-old male patient with liver cancer and portal hypertension received 42 months of co-treatment with atezolizumab and bevacizumab.After that,the disease was rated stable disease.The patient was then diagnosed with gastric cancer.Since the patient was not sensitive to anti-programmed death ligand 1 immunosuppressive agents,a co-treatment with oxaliplatin,tegafur,apatinib,and cadonilimab was selected after multidisciplinary consultation and the patient’s agreement.After four cycles of treatment,partial response and stable disease were observed in gastric and liver cancers,respectively.Surgical treatment was performed considering the high-risk factors of gastrointestinal bleeding in patients with gastroesophageal varices.Postoperative pathology showed that the Tumor Regression Grade was 1.Moreover,the genetic testing of postoperative tumor specimens indicated negative programmed death ligand 1 and microsatellite stability.In addition,the latest follow-up indicated an 8 and 40-month progression-free survival in gastric and liver cancer patients,respectively.Currently,the patient is receiving postoperative immunotherapy with cadonilimab.CONCLUSION Cadonilimab not only treats microsatellite stability gastric cancer patients but can also be used for liver cancer treatment.展开更多
BACKGROUND The introduction of pegaspargase has greatly advanced the treatment of acute lymphoblastic leukemia(ALL).In the literature,only one case of pegaspargaseinduced multiple organ failure has been reported,and t...BACKGROUND The introduction of pegaspargase has greatly advanced the treatment of acute lymphoblastic leukemia(ALL).In the literature,only one case of pegaspargaseinduced multiple organ failure has been reported,and the patient died due to multiple organ failure.CASE SUMMARY Herein,we present a rare case of a 40-year-old man with ALL who developed multiple organ failure after treatment with pegaspargase.The patient had two rare phenomena reflecting poor prognosis,including the discrepancy between clinical manifestations and liver function and persistently low alpha-fetoprotein(AFP)levels from subacute liver failure.However,the patient was successfully treated using a multidisciplinary team approach.CONCLUSION This is the first case report of successful treatment of pegaspargase-induced multiple organ failure.The findings emphasize the importance of a multidisciplinary team approach in treating pegaspargase-induced multiple organ failure.展开更多
BACKGROUND Esophageal cancer is a clinically common malignant tumor of the digestive sys-tem.In 2022,it ranked fifth among the leading causes of cancer-related deaths in China.Its predominant symptom is dysphagia,and ...BACKGROUND Esophageal cancer is a clinically common malignant tumor of the digestive sys-tem.In 2022,it ranked fifth among the leading causes of cancer-related deaths in China.Its predominant symptom is dysphagia,and approximately 30%–40%of patients are prone to developing postoperative recurrent stenosis,necessitating repeated esophageal dilation,which significantly affects patients’quality of life.The self-dilation technique,performed by patients,enables preventive esophageal dilation and aims to reduce the frequency of recurrent stenosis.CASE SUMMARY We report the case of a 61-year-old man who underwent repeated esophageal di-lations following endoscopic submucosal dissection.During his eighth hospital admission,a multidisciplinary management team was established to implement an evidence-based self-help balloon dilation technique,facilitate early identifi-cation of nursing concerns and complications,and provide transitional care fo-llowing discharge.The patient reported a high level of satisfaction during the hospital stay.During the 6-month follow-up after discharge,the patient’s quality of life improved,with a substantial reduction in dysphagia.The esophageal stric-ture was successfully dilated from 5 mm to 6 mm,the interval between readmi-ssions was prolonged,and the patient’s weight increased from 49 kg to 50 kg.CONCLUSION The establishment of a multidisciplinary case management team,combined with the implementation of a self-help balloon dilation technique,early identification and management of nursing issues and complications,and person-alized extended care,can significantly enhance patient satisfaction during hospitalization,improve quality of life,and extend the interval between readmissions.These strategies can provide valuable practical guidance for the clinical treatment and nursing of patients with recurrent esophageal stenosis.展开更多
BACKGROUND Emphysematous gastritis(EG)is a rare and serious condition that has fatal consequences.Although its clinical presentation is not specific,radiological imaging is characterized by intramural gastric gas.Defe...BACKGROUND Emphysematous gastritis(EG)is a rare and serious condition that has fatal consequences.Although its clinical presentation is not specific,radiological imaging is characterized by intramural gastric gas.Defects in the stomach mucosal barrier and invasion of gas-producing organisms are believed to be the cause.CASE SUMMARY An 88-year-old male with multiple comorbidities presented to our center with abdominal pain and increased stoma output as chief complaints.Upon further investigation he was found to have EG.Despite the high mortality risk without intervention,the patient and family declined operative intervention.CONCLUSION This case report underscored the challenges of managing a critically ill elderly patient with a history of multiple comorbidities and extensive abdominal surgeries and highlighted the successful use of conservative measures in treating EG.展开更多
BACKGROUND Multiple primary carcinoma(MPC)refers to two or more types of primary malignant tumors occurring simultaneously or sequentially in the same patient.Breast cancer is one of the most common malignant tumors a...BACKGROUND Multiple primary carcinoma(MPC)refers to two or more types of primary malignant tumors occurring simultaneously or sequentially in the same patient.Breast cancer is one of the most common malignant tumors affecting women.On the other hand,diffuse large B-cell lymphoma(DLBCL)is the most frequent form of non-Hodgkin’s lymphoma(NHL).In clinical practice,the simultaneous existence of metachronous primary breast cancer and lymphoma is rare.In this case,we highlight the significance of multidisciplinary management and advanced imaging techniques in the early identification and treatment of MPC cases.CASE SUMMARY In this study,we report a case of a 40-year-old female who was diagnosed with invasive ductal carcinoma of the breast(T3N1M0 stage IIIA LuminalB type)as the first primary cancer and DLBCL(stage IIIA)as the second primary cancer.The patient underwent the modified radical mastectomy for left breast cancer and received Rituximab,cyclophospha-mide,hydroxydaunorubicin,Oncovin(vincristine)and prednisolone regimen chemotherapy treatment for DLBCL.As of now,the patient is in stable condition.The successful diagnosis of the present patient highlights the need for multidisciplinary management and adoption of advanced imaging techniques to identify the second primary cancer,especially NHL.CONCLUSION Accurate diagnosis and management of metachronous MPC requires an interdisciplinary team and selection of an appropriate treatment plan.展开更多
BACKGROUND Cardiac sarcoidosis(CS)is an infiltrative disease with manifestations such as nonsustained ventricular tachycardia(NSVT)and heart failure(HF).Antiphospholipid syndrome(APS)and antiphospholipid positivity(AP...BACKGROUND Cardiac sarcoidosis(CS)is an infiltrative disease with manifestations such as nonsustained ventricular tachycardia(NSVT)and heart failure(HF).Antiphospholipid syndrome(APS)and antiphospholipid positivity(APP)are prothrombotic phenomena which elevate risk for thromboembolism.CS with active systemic sarcoid and APS/APP is a rare combination of diseases.CASE SUMMARY A 54 year old male with HF presented with several cardiopulmonary symptoms.Chest imaging showed bilateral patchy and reticulonodular infiltrates.Subsequent lung biopsy confirmed pulmonary sarcoidosis.Positron emission tomography revealed active systemic sarcoidosis(SS)and fibrotic CS.Positive antiphospholipid antibodies without thromboembolism confirmed APP.HF and APP were managed with medical therapy.Fibrotic CS and NSVT required permanent cardiac device and antiarrhythmic therapy.SS was managed with early taper of steroids and transition to biologics.CONCLUSION Fibrotic CS with active SS and APS/APP has not been previously described in literature.This case utilized a modified approach for the management of this combination of diseases.As immunosuppressants such as steroids have limited utility in fibrotic sarcoidosis and a potential for thromboembolic complications in the presence of APP,an accelerated transition to non-thrombotic immunosuppressants can be advantageous in the long term treatment of this combination of diseases.展开更多
BACKGROUND Metachronous multiple esophageal squamous cell carcinomas(ESCCs)may occur in some patients after endoscopic resection.Multiple dysplastic lesions in the esophagus increase risk of multiple squamous cell car...BACKGROUND Metachronous multiple esophageal squamous cell carcinomas(ESCCs)may occur in some patients after endoscopic resection.Multiple dysplastic lesions in the esophagus increase risk of multiple squamous cell carcinomas(SCCs).Endoscopic imaging technology such as narrow band imaging(NBI),can detect early SCC.Lugol chromoendoscopy is also the conventional standard technique for detecting superficial ESCC.However,little is known about the interval from the first SCC to the metachronous SCC.Effective methods to prevent multiple metachronous SCCs are needed in survivors of esophageal SCC.CASE SUMMARY A 56-year-old man showed a slightly elevated reddish area in the middle thoracic esophagus at 30 cm from the incisors on gastroscopy for routine examination.Esophageal mucosa lesion was about 2.5 cm.NBI and magnifying gastroscopy confirmed intra-epithelial papillary loop type B-1 according to the Japan Esophageal Society Classification.Lugol chromoendoscopy was used to evaluate the dysplastic squamous epithelium in the esophagus.Biopsy pathology revealed severe dysplastic squamous epithelium.Computed tomography showed no lymph node metastasis.His complete blood test and tumor markers were within reference values.He had no history of alcohol consumption and smoking.Mucosal lesion was dissected by endoscopic submucosal dissection(ESD).Postoperative pathological results showed moderately differentiated squamous carcinoma.No cancer thrombus was seen in the vasculature,and the surrounding cut edge was not involved.The patient underwent radiotherapy within 2 months after ESD.The multiple Lugol-voiding lesions disappeared,and enhanced chest computed tomography revealed no lymph node metastasis.CONCLUSION This is the first case of multiple dysplastic lesions of esophagus cured by radiotherapy.Radiotherapy after minimally invasive endoscopic treatment might be a safe and effective optional therapeutic strategy to prevent metachronous multiple esophageal SCCs.展开更多
BACKGROUND Protein-losing enteropathy(PLE)is a rare cause of hypoalbuminemia that can be attributed to intestinal lymphangiectasia.Patients with Noonan syndrome may present with disorder of lymph vessel formation.Howe...BACKGROUND Protein-losing enteropathy(PLE)is a rare cause of hypoalbuminemia that can be attributed to intestinal lymphangiectasia.Patients with Noonan syndrome may present with disorder of lymph vessel formation.However,PLE is rarely reported with Noonan syndrome.CASE SUMMARY A 15-year-old female was hospitalized multiple times for recurrent edema and diarrhea secondary to hypoalbuminemia.Additional manifestations included a ventricular septal defect at birth,intermuscular hemangioma,slightly wide interocular and intermammary distances,and absence of the distal phalanx of the left little finger since birth.Abdominal computed tomography revealed cavernous transformation of the portal vein,and liver biopsy indicated“porto-sinusoidal vascular disease”.Whole exome and Sanger sequencing revealed a heterozygous mutation(exon9:C.850C>T:P.R284C)in leucine zipper-like transcription regulator 1,suggesting Noonan syndrome type 10.Further examinations revealed thoracic duct dysplasia and intestinal lymphangiectasia causing PLE in this patient.A multidisciplinary team decided to address thoracic duct dysplasia with outlet obstruction.Approximately two years after the microsurgical relief of the thoracic duct outlet obstruction,the patient achieved persistent normal serum albumin level without edema or diarrhea.Furthermore,the relevant literatures on Noonan syndrome and PLE were reviewed.CONCLUSION Herein,we reported the first case of PLE associated with Noonan syndrome caused by a rare genetic mutation in leucine zipper-like transcription regulator 1(c.850C>T:P.R284C)with newly reported manifestations.This case presented the successful treatment of clinical hypoalbuminemia attributed to thoracic duct dysplasia,intestinal lymphangiectasia and PLE.展开更多
AIM:To report and analyze cases of sterile intraocular inflammation(IOI)following intravitreal faricimab injections in patients treated for neovascular age-related macular degeneration(nAMD)and diabetic macular edema(...AIM:To report and analyze cases of sterile intraocular inflammation(IOI)following intravitreal faricimab injections in patients treated for neovascular age-related macular degeneration(nAMD)and diabetic macular edema(DME).METHODS:This double-center case series included nine eyes of six patients who developed uveitis after faricimab therapy.Comprehensive clinical evaluation was performed,including slit-lamp examination,intraocular pressure(IOP)measurement,fluorescein and indocyanine green angiography(ICGA),and laboratory tests.Inflammatory responses were treated with topical or systemic corticosteroids,and patients were monitored for visual acuity and inflammatory activity.RESULTS:The incidence of IOI was 0.8%per patient(Innsbruck)and 0.23%(Czechia),with inflammation typically occurring between the third and sixth injection(mean interval:10d post-injection).Inflammator y presentations ranged from anterior uveitis to posterior segment involvement.One notable case demonstrated novel choroidal hypofluorescent lesions on angiography,suggesting deeper ocular involvement.The mean patient age was 76y;five of six affected patients were female.All cases responded to local and systemic corticosteroids,with full recovery of initial visual acuity.CONCLUSION:Sterile IOI after faricimab appears to be a rare but relevant adverse event.Although the incidence falls within expected ranges for anti-vascular endothelial growth factor(anti-VEGF)agents,the observed choroidal involvement represents a potentially new safety signal.Prompt diagnosis and corticosteroid therapy are effective in all cases.Our findings support the need for vigilant post-marketing surveillance and further studies to better understand the underlying mechanisms and risk factors of faricimab-associated inflammation.展开更多
BACKGROUND Congenital hypothyroidism(CH)is a common condition in both preterm and term infants characterized by either thyroid gland absence or hypofunctionality.The clinical association of refractory lactic acidosis ...BACKGROUND Congenital hypothyroidism(CH)is a common condition in both preterm and term infants characterized by either thyroid gland absence or hypofunctionality.The clinical association of refractory lactic acidosis and heart failure has rarely been observed in cases of pediatric patients with CH pathology.Here,we explored the etiological relationship between CH,heart failure,and refractory lactic acidosis to reflect the importance of thyroid function screening in neonates with heart disease.CASE SUMMARY A 33-day-old extremely premature female infant presented with tachypnea,respiratory distress,recurrent infections,and abdominal distension postnatal.On admission to our facility,she had cardiomegaly,hepatomegaly,and lactic acidosis(revealed on blood gas analysis),with lactate progressively rising to 25 mmol/L.Chest radiographs showed pulmonary congestion,while echocardiography revealed cardiac enlargement,left ventricular wall thickening,and pericardial effusion.Initial management aimed at correcting acidosis and treating heart failure proved ineffective.After reassessment,thyroid function tests showed significantly decreased triiodothyronine,free triiodothyronine,thyroxine,and free thyroxine levels,with a significantly increased thyroidstimulating hormone level,confirming a CH diagnosis.Levothyroxine was administered,resulting in rapid correction of lactic acidosis and gradual improvement of thyroid function and systemic symptoms,culminating in full recovery and discharge.We also reviewed the relevant literature on thyroid and cardiac dysfunctions in order to explore their deeper association.CONCLUSION This case links CH-induced heart failure with refractory lactic acidosis,urging prompt thyroid screening in affected neonates to reduce mortality.展开更多
BACKGROUND SMARCB1/INI1-deficient pancreatic undifferentiated rhabdoid carcinoma is a highly aggressive tumor,and spontaneous splenic rupture(SSR)as its presenting manifestation is rarely reported among pancreatic mal...BACKGROUND SMARCB1/INI1-deficient pancreatic undifferentiated rhabdoid carcinoma is a highly aggressive tumor,and spontaneous splenic rupture(SSR)as its presenting manifestation is rarely reported among pancreatic malignancies.CASE SUMMARY We herein report a rare case of a 59-year-old female who presented with acute left upper quadrant abdominal pain without any history of trauma.Abdominal imaging demonstrated a heterogeneous splenic lesion with hemoperitoneum,raising clinical suspicion of SSR.Emergency laparotomy revealed a pancreatic tumor invading the spleen and left kidney,with associated splenic rupture and dense adhesions,necessitating en bloc resection of the distal pancreas,spleen,and left kidney.Histopathology revealed a biphasic malignancy composed of moderately differentiated pancreatic ductal adenocarcinoma and an undifferentiated carcinoma with rhabdoid morphology and loss of SMARCB1 expression.Immunohistochemical analysis confirmed complete loss of SMARCB1/INI1 in the undifferentiated component,along with a high Ki-67 index(approximately 80%)and CD10 positivity.The ductal adenocarcinoma component retained SMARCB1/INI1 expression and was positive for CK7 and CK-pan.Transitional zones between the two tumor components suggested progressive dedifferentiation and underlying genomic instability.The patient received adjuvant chemotherapy with gemcitabine and nab-paclitaxel and maintained a satisfactory quality of life at the 6-month follow-up.CONCLUSION This study reports a rare case of SMARCB1/INI1-deficient undifferentiated rhabdoid carcinoma of the pancreas combined with ductal adenocarcinoma,presenting as SSR-an exceptionally uncommon initial manifestation of pancreatic malignancy.展开更多
基金Supported by Gansu Provincial Natural Science Foundation,No.21JR1RA010In-Hospital Research Fund of Gansu Provincial Hospital,No.23GSSYD-5.
文摘BACKGROUND The incidence of malignant gastrointestinal(GI)tumors is increasing,and advancements in medical care have significantly improved patient survival rates.As a result,the number of cases involving multiple primary cancers(MPC)has also increased.The rarity of MPC and the absence of sensitive and specific dia-gnostic markers often lead to missed or incorrect diagnoses.It is,therefore,of vital importance to improve the vigilance of clinicians and the accurate diagnosis of this disease.Patients with GI malignancies face a higher relative risk of deve-loping additional primary malignant tumors compared to those with other systemic tumors.Vigilant monitoring and follow-up are crucial,especially for high-risk groups,which include older adults,men,those with addictions to alcohol and tobacco,those with a family history of tumors,and those who have undergone radiotherapy.CASE SUMMARY In this article,we report three cases of MPC,each involving malignant tumors of the GI tract as the initial primary carcinoma,offering insights that may aid in effectively managing similar cases.CONCLUSION Patients with GI malignancies face a higher MPC risk.Developing screening and follow-up protocols may enhance detection and treatment outcomes.
文摘BACKGROUND The liver is the most common site of digestive system tumor metastasis,but not all liver metastases can be traced back to the primary lesions.Although it is unusual,syphilis can impact the liver,manifesting as syphilitic hepatitis with inflammatory nodules,which might be misdiagnosed as metastasis.CASE SUMMARY This case report involves a 46-year-old female who developed right upper abdominal pain and intermittent low fever that persisted for more than three months.No definitive diagnosis of a tumor had been made in the past decades,but signs of multiple liver metastases were recognized after a computed tomo-graphy scan without evidence of primary lesions.With positive serological tests for syphilis and a biopsy of the liver nodules,a diagnosis of hepatic syphilis was made and confirmed with follow-up nodule reduction after anti-syphilis therapy.CONCLUSION Clinicians must be aware of the possibility that syphilis can cause hepatic inflam-matory masses,especially when liver metastasis is suspected without evidence of primary lesions.A definitive diagnosis should be established in conjunction with a review of the patient’s medical history for accurate therapeutic intervention.
文摘BACKGROUND Jejunal diverticula(JD)are rare clinical conditions that are typically incidentally detected and asymptomatic.When acute complications arise,surgical exploration may be necessary for accurate diagnosis and appropriate treatment.In this report,we present a case of multiple JD complicated by gastrointestinal bleeding and review the pathogenesis,diagnosis,and treatment of JD to increase clinician awareness of this condition.CASE SUMMARY A 70-year-old male patient with multiple JD presented with repeated massive gastrointestinal bleeding.The patient did not respond to symptomatic conservative treatment.Additional diagnostic investigations,including digestive endoscopy and abdominal angiography,did not reveal any relevant abnormalities.An exploratory laparotomy was subsequently performed,during which a segment of the bowel containing numerous diverticulum-like structures was surgically removed.Following successful discharge from the hospital,the patient did not experience any further episodes of gastrointestinal bleeding during subsequent follow-up.CONCLUSION Complications caused by JD are often difficult to diagnose,and surgical exploration is sometimes the most appropriate method.
基金Supported by the Zhejiang Medical Science and Technology Project,No.2022KY1325 and No.2023KY381Public Welfare Project of Jinhua Science and Technology Plan,No.2023-4-084Major Project of Jinhua Science and Technology Plan,No.2023-3-066.
文摘BACKGROUND Ganglioneuroma is a rare,well-differentiated,slow-growing benign tumor of the peripheral nerves,with surgical resection being the only curative treatment.Surgical resection of ganglioneuromas encasing major blood vessels remains a substantial clinical challenge.Traditionally,these cases often require open abdominal surgery or combined organ resections,and in some instances,the tumors are considered unresectable.Currently,no reports have described the resection of such tumors via laparoscopy.CASE SUMMARY A 35-year-old woman was admitted to our hospital after the incidental discovery of a retroperitoneal space-occupying lesion.Imaging revealed a mass with the celiac axis and superior mesenteric artery passing through it.A neurogenic tumor was suspected,with ganglioneuroma being the most likely diagnosis.Following comprehensive preoperative preparation,the retroperitoneal tumor was resected using a three-dimensional laparoscopy combined with an organ suspension technique.The surgical approach involved incising the tumor along the vascular axis and conducting meticulous,vascular-preserving tumor excision.The operation lasted approximately 458 minutes,with an estimated blood loss of 50 mL.The patient was discharged on the 8th postoperative day.A transient liver injury occurred after surgery but improved rapidly.After 11 months of postoperative follow-up,no complications or tumor recurrence were observed.CONCLUSION This case illustrates the feasibility of minimally invasive laparoscopic resection for retroperitoneal ganglioneuromas encasing major blood vessels.
文摘BACKGROUND Gastric bezoars are indigestible masses that can lead to gastrointestinal ob-struction and ulceration.Standard treatments include endoscopic mechanical lithotripsy with a polypectomy snare and Coca-Cola dissolution therapy or a combination of both approaches.However,giant bezoars frequently require multiple treatment sessions and extended hospital stays.Additionally,snare-based mechanical fragmentation may be limited by factors such as bezoar size,shape,density,slipperiness,and restricted working space.In cases where refra-ctory giant bezoars are unresponsive to traditional methods,surgical intervention is often necessary.CASE SUMMARY A 57-year-old male with a history of type 2 diabetes presented with severe epigastric pain and vomiting.Endoscopy revealed two large phytobezoars and a gastric ulcer.Initial attempts at mechanical fragmentation with a polypectomy snare and Coca-Cola ingestion for dissolution were unsuccessful due to the large size and complex structure of the bezoars.An innovative approach using snare-tip electrocautery was then employed.It successfully penetrated the slippery,hard surface of the bezoars and fragmented them into smaller pieces.The patient was subsequently treated with Coca-Cola ingestion,enzyme supplements,and proton pump inhibitors.He was discharged without complications following the endoscopic sessions.CONCLUSION Snare-tip electrocautery is a safe,cost-effective,and minimally invasive alter-native for managing large,refractory gastric bezoars.This is a valuable option in resource-limited settings.
基金Wu Jieping Medical Foundation,No.320.6750.2021-02-113.
文摘BACKGROUND Gastric neuroendocrine carcinomas(NECs)exhibit aggressive features,such as rapid growth,higher rate of metastasis,and a generally unfavorable prognosis compared to gastric adenocarcinoma.As a result,therapeutic options for NECs remain limited,contributing to the poor prognosis of patients.Immunotherapy has emerged as a promising treatment strategy and demonstrated the potential to partially improve the survival and prognosis of patients with NECs.Nevertheless,the unique clinical response termed pseudoprogression(PsP)has garnered considerable attention in the context of immunotherapy.CASE SUMMARY Presented here is a case of NEC recurrence five and a half months after radical gastric surgery.The 45-year-old male patient underwent combination treatment involving a PD-1 blocker and tyrosine kinase inhibitors and encountered two instances of PsP during treatment.The patient ultimately achieved a durable treatment response without altering his treatment regimens,resulting in a substantial therapeutic benefit.CONCLUSION This case report aimed to provide the authors’experience with the diagnosis of PsP and treatment strategies for PsP in ongoing immunotherapy.
基金Supported by University Hospital of Giessen and Marburg(UKGM)-Justus-Liebig-University(JLU)-Cooperation Grant,No.7/2016 GIthe Else Kröner-Fresenius-Stiftung,No.2016_A90and the German Research Foundation,No.544054869.
文摘BACKGROUND Here,we report a case of jejunal diverticulosis from an anatomical and histological view.During the“Gross Anatomy course,”we found multiple jejunal diverticula along a total length of 208 cm of intestine.CASE SUMMARY After opening the intestinal tract,we counted 232 jejunal diverticulum entry points with a diameter of up to 2 cm and observed connections between the diverticula that created shortcuts between two distinct intestinal parts.Interestingly,we observed an extreme longitudinal striation on the intestinal parts hosting diverticula.Thorough vessel preparation utilizing a dissecting microscope confirmed that all investigated arteriae rectae ended in a diverticulum.Histological and immunohistochemical investigations revealed that intestinal villi of diverticula were smaller and less prominent than control tissue and that the stratum longitudinale,as well as the stratum circular,were much thinner in the diverticula compared to control tissue.Neither submucosal nor mesenteric plexus could be detected in the diverticula.However,vasoactive intestinal peptidepositive nerve fibers and villin-positive brush border could only be detected in control tissue.This indicates that jejunal diverticulosis is associated with abnormalities of the smooth muscles and a disorder of innervation.CONCLUSION Jejunal diverticulosis originates from mesenteric vessels,featuring smooth muscle changes,absent innervation,and thinning of tissue layers.
基金Supported by STI2030-Major Projects,No.2021ZD0202001Capital Funds for Health Improvement and Research,No.CFH 2022-2-4012.
文摘BACKGROUND Systemic lupus erythematosus(SLE)can affect multiple organs or systems.The involvement of the central nervous system can result in the manifestation of epilepsy,an acute confusional state,and other rare neuropsychiatric presentations,such as catatonia.CASE SUMMARY We present a case of an adolescent male patient with first-onset SLE who presented with neuropsychiatric symptoms including epilepsy and delirium.The initial utilization of olanzapine to alleviate symptoms of agitation precipitated the emergence of catatonia,which was mitigated by discontinuing olanzapine and supplementing with lorazepam.In this case,whether the catatonia was secondary to the utilization of antipsychotics or to an organic disease is a question that warrants differential diagnosis.CONCLUSION Multidisciplinary collaborative management is the cornerstone for the successful management of severe cases of SLE.
文摘BACKGROUND Multiple primary malignant tumors refer to the occurrence of two or more primary malignant tumors in the same organ or multiple organs or tissues at the same time or successively in the same patient,and can occur anywhere in the body.The treatment guidelines for patients with multiple primary malignant tumors are currently controversial.CASE SUMMARY A 51-year-old male patient with liver cancer and portal hypertension received 42 months of co-treatment with atezolizumab and bevacizumab.After that,the disease was rated stable disease.The patient was then diagnosed with gastric cancer.Since the patient was not sensitive to anti-programmed death ligand 1 immunosuppressive agents,a co-treatment with oxaliplatin,tegafur,apatinib,and cadonilimab was selected after multidisciplinary consultation and the patient’s agreement.After four cycles of treatment,partial response and stable disease were observed in gastric and liver cancers,respectively.Surgical treatment was performed considering the high-risk factors of gastrointestinal bleeding in patients with gastroesophageal varices.Postoperative pathology showed that the Tumor Regression Grade was 1.Moreover,the genetic testing of postoperative tumor specimens indicated negative programmed death ligand 1 and microsatellite stability.In addition,the latest follow-up indicated an 8 and 40-month progression-free survival in gastric and liver cancer patients,respectively.Currently,the patient is receiving postoperative immunotherapy with cadonilimab.CONCLUSION Cadonilimab not only treats microsatellite stability gastric cancer patients but can also be used for liver cancer treatment.
基金Supported by National Natural Science Foundation of China,No.82100625Zhejiang Provincial natural Science Foundation of China,No.Q20H030013.
文摘BACKGROUND The introduction of pegaspargase has greatly advanced the treatment of acute lymphoblastic leukemia(ALL).In the literature,only one case of pegaspargaseinduced multiple organ failure has been reported,and the patient died due to multiple organ failure.CASE SUMMARY Herein,we present a rare case of a 40-year-old man with ALL who developed multiple organ failure after treatment with pegaspargase.The patient had two rare phenomena reflecting poor prognosis,including the discrepancy between clinical manifestations and liver function and persistently low alpha-fetoprotein(AFP)levels from subacute liver failure.However,the patient was successfully treated using a multidisciplinary team approach.CONCLUSION This is the first case report of successful treatment of pegaspargase-induced multiple organ failure.The findings emphasize the importance of a multidisciplinary team approach in treating pegaspargase-induced multiple organ failure.
文摘BACKGROUND Esophageal cancer is a clinically common malignant tumor of the digestive sys-tem.In 2022,it ranked fifth among the leading causes of cancer-related deaths in China.Its predominant symptom is dysphagia,and approximately 30%–40%of patients are prone to developing postoperative recurrent stenosis,necessitating repeated esophageal dilation,which significantly affects patients’quality of life.The self-dilation technique,performed by patients,enables preventive esophageal dilation and aims to reduce the frequency of recurrent stenosis.CASE SUMMARY We report the case of a 61-year-old man who underwent repeated esophageal di-lations following endoscopic submucosal dissection.During his eighth hospital admission,a multidisciplinary management team was established to implement an evidence-based self-help balloon dilation technique,facilitate early identifi-cation of nursing concerns and complications,and provide transitional care fo-llowing discharge.The patient reported a high level of satisfaction during the hospital stay.During the 6-month follow-up after discharge,the patient’s quality of life improved,with a substantial reduction in dysphagia.The esophageal stric-ture was successfully dilated from 5 mm to 6 mm,the interval between readmi-ssions was prolonged,and the patient’s weight increased from 49 kg to 50 kg.CONCLUSION The establishment of a multidisciplinary case management team,combined with the implementation of a self-help balloon dilation technique,early identification and management of nursing issues and complications,and person-alized extended care,can significantly enhance patient satisfaction during hospitalization,improve quality of life,and extend the interval between readmissions.These strategies can provide valuable practical guidance for the clinical treatment and nursing of patients with recurrent esophageal stenosis.
文摘BACKGROUND Emphysematous gastritis(EG)is a rare and serious condition that has fatal consequences.Although its clinical presentation is not specific,radiological imaging is characterized by intramural gastric gas.Defects in the stomach mucosal barrier and invasion of gas-producing organisms are believed to be the cause.CASE SUMMARY An 88-year-old male with multiple comorbidities presented to our center with abdominal pain and increased stoma output as chief complaints.Upon further investigation he was found to have EG.Despite the high mortality risk without intervention,the patient and family declined operative intervention.CONCLUSION This case report underscored the challenges of managing a critically ill elderly patient with a history of multiple comorbidities and extensive abdominal surgeries and highlighted the successful use of conservative measures in treating EG.
文摘BACKGROUND Multiple primary carcinoma(MPC)refers to two or more types of primary malignant tumors occurring simultaneously or sequentially in the same patient.Breast cancer is one of the most common malignant tumors affecting women.On the other hand,diffuse large B-cell lymphoma(DLBCL)is the most frequent form of non-Hodgkin’s lymphoma(NHL).In clinical practice,the simultaneous existence of metachronous primary breast cancer and lymphoma is rare.In this case,we highlight the significance of multidisciplinary management and advanced imaging techniques in the early identification and treatment of MPC cases.CASE SUMMARY In this study,we report a case of a 40-year-old female who was diagnosed with invasive ductal carcinoma of the breast(T3N1M0 stage IIIA LuminalB type)as the first primary cancer and DLBCL(stage IIIA)as the second primary cancer.The patient underwent the modified radical mastectomy for left breast cancer and received Rituximab,cyclophospha-mide,hydroxydaunorubicin,Oncovin(vincristine)and prednisolone regimen chemotherapy treatment for DLBCL.As of now,the patient is in stable condition.The successful diagnosis of the present patient highlights the need for multidisciplinary management and adoption of advanced imaging techniques to identify the second primary cancer,especially NHL.CONCLUSION Accurate diagnosis and management of metachronous MPC requires an interdisciplinary team and selection of an appropriate treatment plan.
文摘BACKGROUND Cardiac sarcoidosis(CS)is an infiltrative disease with manifestations such as nonsustained ventricular tachycardia(NSVT)and heart failure(HF).Antiphospholipid syndrome(APS)and antiphospholipid positivity(APP)are prothrombotic phenomena which elevate risk for thromboembolism.CS with active systemic sarcoid and APS/APP is a rare combination of diseases.CASE SUMMARY A 54 year old male with HF presented with several cardiopulmonary symptoms.Chest imaging showed bilateral patchy and reticulonodular infiltrates.Subsequent lung biopsy confirmed pulmonary sarcoidosis.Positron emission tomography revealed active systemic sarcoidosis(SS)and fibrotic CS.Positive antiphospholipid antibodies without thromboembolism confirmed APP.HF and APP were managed with medical therapy.Fibrotic CS and NSVT required permanent cardiac device and antiarrhythmic therapy.SS was managed with early taper of steroids and transition to biologics.CONCLUSION Fibrotic CS with active SS and APS/APP has not been previously described in literature.This case utilized a modified approach for the management of this combination of diseases.As immunosuppressants such as steroids have limited utility in fibrotic sarcoidosis and a potential for thromboembolic complications in the presence of APP,an accelerated transition to non-thrombotic immunosuppressants can be advantageous in the long term treatment of this combination of diseases.
基金Supported by Jinhua City Science and Technology Plan Projects,No.2022-4-145.
文摘BACKGROUND Metachronous multiple esophageal squamous cell carcinomas(ESCCs)may occur in some patients after endoscopic resection.Multiple dysplastic lesions in the esophagus increase risk of multiple squamous cell carcinomas(SCCs).Endoscopic imaging technology such as narrow band imaging(NBI),can detect early SCC.Lugol chromoendoscopy is also the conventional standard technique for detecting superficial ESCC.However,little is known about the interval from the first SCC to the metachronous SCC.Effective methods to prevent multiple metachronous SCCs are needed in survivors of esophageal SCC.CASE SUMMARY A 56-year-old man showed a slightly elevated reddish area in the middle thoracic esophagus at 30 cm from the incisors on gastroscopy for routine examination.Esophageal mucosa lesion was about 2.5 cm.NBI and magnifying gastroscopy confirmed intra-epithelial papillary loop type B-1 according to the Japan Esophageal Society Classification.Lugol chromoendoscopy was used to evaluate the dysplastic squamous epithelium in the esophagus.Biopsy pathology revealed severe dysplastic squamous epithelium.Computed tomography showed no lymph node metastasis.His complete blood test and tumor markers were within reference values.He had no history of alcohol consumption and smoking.Mucosal lesion was dissected by endoscopic submucosal dissection(ESD).Postoperative pathological results showed moderately differentiated squamous carcinoma.No cancer thrombus was seen in the vasculature,and the surrounding cut edge was not involved.The patient underwent radiotherapy within 2 months after ESD.The multiple Lugol-voiding lesions disappeared,and enhanced chest computed tomography revealed no lymph node metastasis.CONCLUSION This is the first case of multiple dysplastic lesions of esophagus cured by radiotherapy.Radiotherapy after minimally invasive endoscopic treatment might be a safe and effective optional therapeutic strategy to prevent metachronous multiple esophageal SCCs.
基金Supported by the Shandong Provincial Natural Science Foundation of China,No.ZR2023QH015Qingdao Municipal Natural Science Foundation of China,No.23-2-1-134-zyyd-jch.
文摘BACKGROUND Protein-losing enteropathy(PLE)is a rare cause of hypoalbuminemia that can be attributed to intestinal lymphangiectasia.Patients with Noonan syndrome may present with disorder of lymph vessel formation.However,PLE is rarely reported with Noonan syndrome.CASE SUMMARY A 15-year-old female was hospitalized multiple times for recurrent edema and diarrhea secondary to hypoalbuminemia.Additional manifestations included a ventricular septal defect at birth,intermuscular hemangioma,slightly wide interocular and intermammary distances,and absence of the distal phalanx of the left little finger since birth.Abdominal computed tomography revealed cavernous transformation of the portal vein,and liver biopsy indicated“porto-sinusoidal vascular disease”.Whole exome and Sanger sequencing revealed a heterozygous mutation(exon9:C.850C>T:P.R284C)in leucine zipper-like transcription regulator 1,suggesting Noonan syndrome type 10.Further examinations revealed thoracic duct dysplasia and intestinal lymphangiectasia causing PLE in this patient.A multidisciplinary team decided to address thoracic duct dysplasia with outlet obstruction.Approximately two years after the microsurgical relief of the thoracic duct outlet obstruction,the patient achieved persistent normal serum albumin level without edema or diarrhea.Furthermore,the relevant literatures on Noonan syndrome and PLE were reviewed.CONCLUSION Herein,we reported the first case of PLE associated with Noonan syndrome caused by a rare genetic mutation in leucine zipper-like transcription regulator 1(c.850C>T:P.R284C)with newly reported manifestations.This case presented the successful treatment of clinical hypoalbuminemia attributed to thoracic duct dysplasia,intestinal lymphangiectasia and PLE.
文摘AIM:To report and analyze cases of sterile intraocular inflammation(IOI)following intravitreal faricimab injections in patients treated for neovascular age-related macular degeneration(nAMD)and diabetic macular edema(DME).METHODS:This double-center case series included nine eyes of six patients who developed uveitis after faricimab therapy.Comprehensive clinical evaluation was performed,including slit-lamp examination,intraocular pressure(IOP)measurement,fluorescein and indocyanine green angiography(ICGA),and laboratory tests.Inflammatory responses were treated with topical or systemic corticosteroids,and patients were monitored for visual acuity and inflammatory activity.RESULTS:The incidence of IOI was 0.8%per patient(Innsbruck)and 0.23%(Czechia),with inflammation typically occurring between the third and sixth injection(mean interval:10d post-injection).Inflammator y presentations ranged from anterior uveitis to posterior segment involvement.One notable case demonstrated novel choroidal hypofluorescent lesions on angiography,suggesting deeper ocular involvement.The mean patient age was 76y;five of six affected patients were female.All cases responded to local and systemic corticosteroids,with full recovery of initial visual acuity.CONCLUSION:Sterile IOI after faricimab appears to be a rare but relevant adverse event.Although the incidence falls within expected ranges for anti-vascular endothelial growth factor(anti-VEGF)agents,the observed choroidal involvement represents a potentially new safety signal.Prompt diagnosis and corticosteroid therapy are effective in all cases.Our findings support the need for vigilant post-marketing surveillance and further studies to better understand the underlying mechanisms and risk factors of faricimab-associated inflammation.
文摘BACKGROUND Congenital hypothyroidism(CH)is a common condition in both preterm and term infants characterized by either thyroid gland absence or hypofunctionality.The clinical association of refractory lactic acidosis and heart failure has rarely been observed in cases of pediatric patients with CH pathology.Here,we explored the etiological relationship between CH,heart failure,and refractory lactic acidosis to reflect the importance of thyroid function screening in neonates with heart disease.CASE SUMMARY A 33-day-old extremely premature female infant presented with tachypnea,respiratory distress,recurrent infections,and abdominal distension postnatal.On admission to our facility,she had cardiomegaly,hepatomegaly,and lactic acidosis(revealed on blood gas analysis),with lactate progressively rising to 25 mmol/L.Chest radiographs showed pulmonary congestion,while echocardiography revealed cardiac enlargement,left ventricular wall thickening,and pericardial effusion.Initial management aimed at correcting acidosis and treating heart failure proved ineffective.After reassessment,thyroid function tests showed significantly decreased triiodothyronine,free triiodothyronine,thyroxine,and free thyroxine levels,with a significantly increased thyroidstimulating hormone level,confirming a CH diagnosis.Levothyroxine was administered,resulting in rapid correction of lactic acidosis and gradual improvement of thyroid function and systemic symptoms,culminating in full recovery and discharge.We also reviewed the relevant literature on thyroid and cardiac dysfunctions in order to explore their deeper association.CONCLUSION This case links CH-induced heart failure with refractory lactic acidosis,urging prompt thyroid screening in affected neonates to reduce mortality.
文摘BACKGROUND SMARCB1/INI1-deficient pancreatic undifferentiated rhabdoid carcinoma is a highly aggressive tumor,and spontaneous splenic rupture(SSR)as its presenting manifestation is rarely reported among pancreatic malignancies.CASE SUMMARY We herein report a rare case of a 59-year-old female who presented with acute left upper quadrant abdominal pain without any history of trauma.Abdominal imaging demonstrated a heterogeneous splenic lesion with hemoperitoneum,raising clinical suspicion of SSR.Emergency laparotomy revealed a pancreatic tumor invading the spleen and left kidney,with associated splenic rupture and dense adhesions,necessitating en bloc resection of the distal pancreas,spleen,and left kidney.Histopathology revealed a biphasic malignancy composed of moderately differentiated pancreatic ductal adenocarcinoma and an undifferentiated carcinoma with rhabdoid morphology and loss of SMARCB1 expression.Immunohistochemical analysis confirmed complete loss of SMARCB1/INI1 in the undifferentiated component,along with a high Ki-67 index(approximately 80%)and CD10 positivity.The ductal adenocarcinoma component retained SMARCB1/INI1 expression and was positive for CK7 and CK-pan.Transitional zones between the two tumor components suggested progressive dedifferentiation and underlying genomic instability.The patient received adjuvant chemotherapy with gemcitabine and nab-paclitaxel and maintained a satisfactory quality of life at the 6-month follow-up.CONCLUSION This study reports a rare case of SMARCB1/INI1-deficient undifferentiated rhabdoid carcinoma of the pancreas combined with ductal adenocarcinoma,presenting as SSR-an exceptionally uncommon initial manifestation of pancreatic malignancy.
