Camrelizumab(SHR-1210) leading to reactive capillary hemangioma in the gingiva: A case report

BACKGROUND Oncologic immunotherapy is attracting attention as an effective strategy for cancer treatment. Currently, there are two kinds of inhibitors: Anti-PD-1 antibodies and anti-PD-L1 antibodies. These inhibitors have shown significant implications in improving the outcomes of certain cancer types in recent years.However, along with its effectiveness, adverse events cannot be ignored. As an anti-PD-1 antibody, camrelizumab(SHR-1210) has some side effects in tumor immunotherapy. The most common adverse event is reactive capillary hemangioma. While it is widely reported to occur in the skin, gingival reactive capillary hemangioma is rarely reported.CASE SUMMARY A 54-year-old man complained of gingival overgrowth on the anterior aspect of the maxilla and mandible for more than 6 mo. He had been placed on SHR-1210 for lung cancer for 7 mo. A gingival mass extending from canine to canine was noted on the lingual surfaces of the mandible. Gingival enlargement was noted in the front teeth. A clinical diagnosis of gingival reactive capillary hemangioma and chronic periodontitis was made. The treatment involved a complex local treatment(repeated local applications of an antibiotic paste, scaling and root planning, and surgery). The excised tissue was sent for histopathological examination, which confirmed the diagnosis of capillary hemangioma. After the operation, most of the gingival enlargement was reduced. At the 2-mo follow-up,it was noted that the gingival overgrowth was immediately reduced after the replacement of the anti-PD-1 agent with an anti-PD-L1 agent.CONCLUSION As the prescription for SHR-1210 has increased considerably in recent years, the occurrence of its possible side effects, including gingival reactive capillary hemangioma, has increased. It is recommended that regular oral examinations be performed before and during the treatment of tumors with SHR-1210.

Lobular capillary hemangioma of the liver

BACKGROUND: Lobular capillary hemangioma (LCH) is a benign vascular tumor that is rare in adults and has never been reported in the liver. This vascular lesion usually presents on the skin or mucous membranes, and predominantly affects children. METHODS: LCH as a large asymptomatic hepatic mass was seen in a 35-year-old female. Imaging and pathologic characteristics of the mass are reviewed, and the relevant literature is also reviewed. RESULTS: A large vascular hepatic lesion was observed in an asymptomatic 35-year-old female. Pathologic examination after surgical resection revealed typical features of LCH. CONCLUSIONS: This is the first case of lobular capillary hemangioma seen as a liver lesion in an adult. Large vascular hepatic lesions pose significant difficulties in discerning benign from potentially malignant conditions. In this report we describe the pitfalls and radiological uncertainties with interpreting vascular lesions of the liver. (Hepatobiliary Pancreat Dis Int 2009; 8: 323-325)

Reactive capillary hemangiomas: a novel dermatologic toxicity following anti-PD-1 treatment with SHR-1210

Objective: SHR-1210 is a new and promising anti-PD-1 agent for solid tumors. During the phase I study of SHR-1210, we encountered a novel but prevalent immune-related dermatologic toxicity: reactive capillary hemangiomas(RCHs). Thus we tried to summarize the features of RCHs and estimate their relationship with tumor response.Methods: This prospective observational study systematically enrolled 98 patients with advanced solid tumors from April 27th,2016 to June 8th, 2017 in the context of the phase I clinical study of SHR-1210. This report focused on the skin toxicities. Patients underwent entire skin inspection every two weeks while taking medication. The clinical course of RCHs was recorded and their association with tumor response was estimated. The data cut-off date was November 15th, 2017.Results: After a median follow-up of 242(range, 29–567) days, RCHs were observed in 85.7%(84/98) of patients on cutaneous/mucosal surfaces; 84.5%(71/84) of the RCHs were evaluated as grade 1 adverse events. No grade 3 or 4 RCHs were observed. The time of onset of RCHs was dose dependent and shortest in the 400 mg-dose cohort(P < 0.001). Spontaneous and complete regression of RCHs was observed both during and after treatment. The objective response rate of tumors for patients with RCHs was 28.9%(24/83). However, no responders were observed among the patients without RCHs.Conclusions: RCHs were prevalent but manageable during treatment with SHR-1210. It might add to the expanding literature regarding immune-related dermatologic adverse events.

Selective embolization can effectively alleviate bleeding symptoms in patients with anorectal hemangioma

In this manuscript,I comment on the article by Pospisilova et al published in the recent issue of the journal,in which selective embolization was used to treat anorectal hemangioma,a rare disease causing lower gastrointestinal bleeding.Anorectal hemangioma can easily be mistaken;for example,the patient in this case was previously misdiagnosed with ulcerative colitis.Choosing the appropriate tests and understanding the typical manifestations of anorectal hemangioma under colonoscopy,computerized tomography,magnetic resonance imaging and other tests are beneficial for diagnosis.The patient presented with intermittent rectal bleeding despite treatment with azathioprine and mesalazine and required blood transfusions since the degree of rectal bleeding worsened.Selective embolization successfully alleviated the patient’s bleeding symptoms and avoided the need for repeated blood transfusions.Tranexamic acid may be useful,considering that the patient still has bleeding symptoms and requires parenteral iron supplementation.

Early and accurate diagnosis and selection of appropriate treatment plans are crucial for patients with gastrointestinal hemangiomas

Gastrointestinal hemangioma(GIH)is clinically rare,accounting for 7%-10%of benign gastrointestinal tumors and 0.5%of systemic hemangiomas.GIH can occur as either solitary or multiple lesions,with gastrointestinal bleeding as a significant clinical manifestation.Understanding the clinical and endoscopic features of GIH is essential for improving diagnostic accuracy,particularly through endoscopy and selective arteriography,which are highly effective in diagnosing GIH and preventing misdiagnosis and inappropriate treatment.Upon confirmed diagnosis,it is essential to thoroughly evaluate the patient's condition to determine the most suitable treatment modality—whether surgical,endoscopic,or minimally invasive intervention.The minimally invasive interventional partial embolization therapy using polyvinyl alcohol particles,proposed and implemented by Pospisilova et al,has achieved excellent clinical outcomes.This approach reduces surgical trauma and the inherent risks of traditional surgical treatments.

Hepatic hemangiomas mimicking gastrointestinal stromal tumors: A case report

BACKGROUND Hepatic hemangiomas can be challenging to diagnose,particularly when they present with atypical features that mimic other conditions,such as gastrointestinal stromal tumors(GISTs).This case highlights the diagnostic difficulties encountered when imaging subepithelial lesions,especially when conventional methods such as computed tomography(CT)and endoscopic ultrasound(EUS)are used.CASE SUMMARY A 44-year-old woman presented with intermittent abdominal distension and heartburn for three months.Her medical history included iron deficiency anemia,menorrhagia,and previous cholecystectomy.One week prior to admission,an endoscopy suggested a bulging gastric fundus,which was likely a GIST,along with chronic nonatrophic gastritis and bile reflux.CT and EUS revealed nodules in the gastric fundus,which were initially considered benign tumors with a differential diagnosis of stromal tumor or leiomyoma.During surgery,unexpected lesions were found in the liver pressing against the gastric fundus,leading to laparoscopic liver resection.Postoperative pathology confirmed the diagnosis of hepatic cavernous hemangiomas.The patient recovered well and was discharged five days later,with normal follow-up results at three months.CONCLUSION This case underscores the challenges in the preoperative diagnosis of GISTs,particularly the limitations of the use of CT and EUS for the evaluation of subepithelial lesions.While CT is the primary tool for visualizing abdominal tumors,it is difficult to detect smaller lesions and assess the layers of the gastrointestinal wall on CT.EUS is recommended for the evaluation of nodules smaller than 2 cm and is useful for distinguishing GISTs from other lesions;however,its accuracy with regard to the differential diagnosis is relatively low.In this case,the gastric distension observed on imaging led to the compression of a liver tumor against the stomach,resulting in the misinterpretation of the tumor as a gastric wall lesion.

Microvenular hemangioma located in the breast:a case report

Microvenular hemangioma(MVH)is a rare,benign type of hemangioma that typically presents as a solitary,slow-growing,red-to-violaceous asymptomatic papule,plaque,or nodule.It most commonly locations of the lesions on the limbs and trunk,with no prior reports of MVH confined to the breast.We present the first reported case of multiple MVH confined to the breast in a 17-year-old female patient.The case is notable for its unique and atypical clinical features.Diagnosing MVH is challenging and requires routine pathological and im-munohistochemical staining.Histological testing revealed typical features of MVH,irregularly branched small or medium vascular interstitial spaces lined with benign endothelial cells and positivity for CD34 and CD31.This case emphasizes the need to consider MVH in the dif-ferential diagnosis of breast vascular lesions,particularly in young patients.It also underscores the importance of histological and immu-nohistochemical analyses for an accurate diagnosis.

Capillary Hemangioma of the Middle Ear and External Auditory Canal:a Case Report

We report a case of capillary hemangiomsa that involved the entire middle ear space,external auditory canal(EAC) and tympanic antrum.Symptoms in the case included ear fullness,hearing loss,otalgia and otorrhea.The case was misdiagnosed as recurrent chronic otitis media with granulation preoperatively.A diagnosis of capillary hemangioma was established by postoperative histological examination.The management of capillary hemangioma of the middle ear and external auditory canal is discussed,with a review of the literature.Because of its variable and sometimes misleading clinical presentation,hemangioma can initially be misdiagnosed as other lesions.Therefore,a high index of suspicion is necessary for early and accurate diagnosis.

Renal anastomosing hemangioma following partial nephrectomy for renal cell carcinoma:A case report

BACKGROUND Renal anastomosing hemangioma(AH)is a rare benign vascular tumor characterized by unique histopathological features.CASE SUMMARY We report a highly unusual case of renal AH.A male patient had undergone partial nephrectomy for clear cell carcinoma of the kidney four years prior.A follow-up computed tomography scan in the third postoperative year revealed a new mass near the surgical site on the same side of the kidney,raising suspicions of tumor recurrence.However,the characteristics on contrast-enhanced magnetic resonance imaging and ultrasonography were more consistent with those of a benign lesion.The patient strongly insisted on undergoing surgery due to concerns about the possibility of renal cancer recurrence.Postoperative pathology confirmed the diagnosis of renal AH.CONCLUSION This case report presents the imaging features of a patient with rare renal AH and a history of renal clear cell carcinoma,providing broader insights into the differential diagnosis of new lesions after surgery for renal cell carcinoma.

Capillary Hemangioma of the Nasal Septum: A Rare Case Report

Capillary hemangioma is a congenital malformation of the capillary. It commonly affects the face, eyelids, lips and skin. Adult capillary hemangioma involving the nasal cavity is rare and may be misdiagnosed as polyps or another tumor. This case report illustrated a middle-age adult patient in Malaysia who presents with unilateral nasal obstruction and intermittent epistaxis who later confirmed capillary hemangioma involving the right nasal septum. Endoscopic surgery excision of the hemangioma was successfully performed. There were no known risk factors present in him. A treating doctor in the outpatient clinic seeing symptoms such as unilateral nasal obstruction must consider alternative diagnosis such as hemangioma besides usual diagnosis of polyps.

Retrospective analysis based on a clinical grading system for patients with hepatic hemangioma:A single center experience

BACKGROUND The optimal approach for managing hepatic hemangioma is controversial.AIM To evaluate a clinical grading system for management of hepatic hemangioma based on our 17-year of single institution experience.METHODS A clinical grading system was retrospectively applied to 1171 patients with hepatic hemangioma from January 2002 to December 2018.Patients were classified into four groups based on the clinical grading system and treatment:(1)Observation group with score<4(Obs score<4);(2)Surgical group with score<4(Sur score<4);(3)Observation group with score≥4(Obs score≥4);and(4)Surgical group with score≥4(Sur score≥4).The clinico-pathological index and outcomes were evaluated.RESULTS There were significantly fewer symptomatic patients in surgical groups(Sur score≥4 vs Obs score≥4,P<0.001;Sur score<4 vs Obs score<4,χ^(2)=8.60,P=0.004;Sur score≥4 vs Obs score<4,P<0.001).The patients in Sur score≥4 had a lower rate of in need for intervention and total patients with adverse event than in Obs score≥4(P<0.001;P<0.001).Nevertheless,there was no significant difference in need for intervention and total patients with adverse event between the Sur score<4 and Obs score<4(P>0.05;χ^(2)=1.68,P>0.05).CONCLUSION This clinical grading system appeared as a practical tool for hepatic hemangioma.Surgery can be suggested for patients with a score≥4.For those with<4,follow-up should be proposed.

Misdiagnosis of hemangioma of left triangular ligament of the liver as gastric submucosal stromal tumor:Two case reports

BACKGROUND Extragastric lesions are typically not misdiagnosed as gastric submucosal tumor(SMT).However,we encountered two rare cases where extrinsic lesions were misdiagnosed as gastric SMTs.CASE SUMMARY We describe two cases of gastric SMT-like protrusions initially misdiagnosed as gastric SMTs by the abdominal contrast-enhanced computed tomography(CT)and endoscopic ultrasound(EUS).Based on the CT and EUS findings,the patients underwent gastroscopy;however,no tumor was identified after incising the gastric wall.Subsequent surgical exploration revealed no gastric lesions in both patients,but a mass was found in the left triangular ligament of the liver.The patients underwent laparoscopic tumor resection,and the postoperative diagnosis was hepatic hemangiomas.CONCLUSION During EUS procedures,scanning across different layers and at varying degrees of gastric cavity distension,coupled with meticulous image analysis,has the potential to mitigate the likelihood of such misdiagnoses.

Portal Venous Thrombosis and Splenic Hemangioma, Secondary to Acute Pancreatitis: Case Report

We present an unusual case of portal vein thrombosis with a splanchnic hemangioma secondary to acute biliary pancreatitis. We report a 45-year-old patient, who has systemic arterial hypertension in treatment, was admitted for abdominal pain in the epigastrium, with irradiation to the right hypochondrium, accompanied by nausea and vomiting of 10 occasions of bile content, physical examination with pain in the right hypochondrium, Murphy positive. We have laboratory studies with a lipase of 788, so a diagnosis of pancreatitis is made with an etiology to be determined. The laboratories suggestive of acute biliary pancreatitis (lipase 788.71);an imaging study was subsequently performed (ultrasonography) with the result of stone in the common bile duct. A laparoscopy was performed with relative improvement, so he was discharged and returned 20 days after surgery due to abdominal pain of the same intensity in the left hypochondrium. Ending his hospitalization with a splenectomy for splenic hemangioma with portal vein thrombosis.

Borderline resectable giant hepatic cavernous hemangioma and coexisting hemangiomatosis should be a new indication for living donor liver transplantation:A report of two cases

To the Editor:Hemangioma is a benign liver tumor that rarely requires treatment if the patient is asymptomatic[1].However,great cavernous hemangioma(GCH)can lead to symptoms due to its mass effect and Kasabach-Merritt syndrome(KMS)[2].GCH treatment options vary;therefore,tailoring treatment to individual patients according to their condition,such as symptoms,tumor location,and liver function,is important.Occasionally,GCH is associated with hemangiomatosis,and its boundaries with normal tissue are unclear[3],leading to a lack of consensus on the initial therapeutic approach,with literature primarily comprising case reports or series[4].This study presented two cases of GCH and coexisting hemangiomatosis;the patients underwent liver resection of the main mass to relieve symptoms but ultimately required liver transplantation(LT).We aimed to describe the role of LT in these patients.

Bilateral circumscribed choroidal hemangioma:a case report

Dear Editor,Bilateral circumscribed choroidal hemangioma(CCH)is mostly associated with Sturge-Weber syndrome and rarely occurs in healthy individuals.So,we present the case about bilateral CCH that occurred in a healthy person and to evaluate the efficacy of 0.05 mL of anti-vascular endothelial growth factor(anti-VEGF)medication and laser photocoagulation in the treatment of CCH.We obtained the written informed consent from the patient,and this case study was in accordance with the tenets of the Declaration of Helsinki.

A new interacting capillary bundle model on the multiphase flow in micropores of tight rocks

Surfactants are widely used in the fracturing fluid to enhance the imbibition and thus the oil recovery rate. However, current numerical models cannot capture the physics behind capillary imbibition during the wettability alteration by surfactants. Although the interacting capillary bundle(ICB) model shows potential in characterizing imbibition rates in different pores during wettability alteration, the existing ICB models neglect the influence of wettability and viscosity ratio on the imbibition behavior, making it difficult to accurately describe the oil-water imbibition behavior within the porous media. In this work,a new ICB mathematical model is established by introducing pressure balance without assuming the position of the leading front to comprehensively describe the imbibition behavior in a porous medium under different conditions, including gas-liquid spontaneous imbibition and oil-water imbibition.When the pore size distribution of a tight rock is known, this new model can predict the changes of water saturation during the displacement process in the tight rock, and also determine the imbibition rate in pores of different sizes. The water saturation profiles obtained from the new model are validated against the waterflooding simulation results from the CMG, while the imbibition rates calculated by the model are validated against the experimental observations of gas-liquid spontaneous imbibition. The good match above indicates the newly proposed model can show the water saturation profile at a macroscopic scale while capture the underlying physics of the multiphase flow in a porous medium at a microscopic scale. Simulation results obtained from this model indicate that both wettability and viscosity ratio can affect the sequence of fluid imbibition into pores of different sizes during the multiphase flow, where less-viscous wetting fluid is preferentially imbibed into larger pores while more-viscous wetting fluid tends to be imbibed into smaller pores. Furthermore, this model provides an avenue to calculate the imbibition rate in pores of different sizes during wettability alteration and capture the non-Darcy effect in micro-and nano-scale pores.

One Case of Right Posterior Mediastinum Intraneural Hemangioma Misdiagnosed as Neurilemmoma

Although rare, intraneural hemangiomas should be considered in the differential diagnosis of peripheral nerve lesions. We report on a 59-year-old female patient, who was admitted to the hospital due to the discovery of bilateral breast masses for 3 months, there was no paresthesia or dyskinesia. The patient accidentally found a mass in the right upper mediastinum while completing a plain chest X-ray, initially suspected as a benign neurilemmoma on CT. Surgical resection and pathological analysis confirmed an intraneural hemangioma. Unexpectedly, the patient developed new-onset right upper limb numbness and paresthesia 3 months post-operatively, probably related to surgical nerve injury. This case underscores the importance of maintaining a broad differential for mediastinal masses, and the potential for iatrogenic neurological complications when managing these rare, yet vascular lesions.

Clinical characteristics of renal anastomotic hemangioma

In this editorial,we comment on the article by Chen and Cai.We focus on renal anastomotic hemangioma,which is a rare benign hemangiomatous disease.This disease has unique clinical characteristics.Its biological behavior is benign,but its imaging results are similar to those of renal cancer.Renal anastomotic hemangioma is easy to misdiagnose and can lead to unnecessary radical nephrectomy.Therefore,urologists need a better understanding of this disease.We believe that patients with renal anastomotic hemangioma should receive individualized diagnosis and treatment to avoid overtreatment.

Reconsideration of the clinical management of hepatic hemangioma

In this letter,we comment on the article by Zhou et al that was published in the recent issue of the World Journal of Gastrointestinal Surgery.This article proposes a new clinical grading system based on a multidisciplinary team,which prompts us to rethink the clinical management of hepatic hemangioma.Hepatic hemangioma is the most common benign solid liver tumor.In general,follow-up and obser-vation for the vast majority of hepatic hemangioma is reasonable.For those pa-tients with symptoms and severe complications,surgical intervention is ne-cessary.Specific surgical indications,however,are still not clear.An effective grading system is helpful in further guiding the clinical management of hepatic hemangioma.In this article,we review the recent literature,summarize the sur-gical indications and treatment of hepatic hemangioma,and evaluate the potential of this new clinical grading system.

Endoscopic ultrasound-guided lauromacrogol injection for treatment of colorectal cavernous hemangioma:Two case reports

BACKGROUND Colorectal cavernous hemangioma is a rare vascular malformation resulting in recurrent lower gastrointestinal hemorrhage,and can be misinterpreted as colitis.Surgical resection is currently the mainstay of treatment,with an emphasis on sphincter preservation.CASE SUMMARY We present details of two young patients with a history of persistent hematochezia diagnosed with colorectal cavernous hemangioma by endoscopic ultrasound(EUS).Cavernous hemangioma was relieved by several EUS-guided lauromacrogol injections and the patients achieved favorable clinical prognosis.CONCLUSION Multiple sequential EUS-guided injections of lauromacrogol is a safe,effective,cost-efficient,and minimally invasive alternative for colorectal cavernous hemangioma.