Immunotherapy-related pneumonitis and bacterial pneumonia after the successful treatment of metastatic malignant melanoma with pembrolizumab: A case report

Introduction:Pembrolizumab, a monoclonal antibody targeting programmed cell death-1 (PD-1), is approved as a therapy for unresectable or metastatic melanoma. Immunotherapy-associated pneumonitis is an uncommon event.Patient concerns:A 73-year-old man was admitted to our hospital with a history of melanoma on the left side of the face (resected in December 2012) and metastasis to the left lung upper lobe (resected in November 2016). Recurrence of metastasis to the bilateral lungs and left pleura was detected in April 2018. A complete response was achieved following treatment with pembrolizumab, with lower limb rashes the only adverse events occurring during therapy. The patient was readmitted in March 2019 with a productive cough, shortness of breath, and mild fever, and sputum culture identified Escherichia coli.Diagnosis:A diagnosis of pneumonia was made, and although cough and shortness of breath responded to ceftazidime and levofloxacin, but fever and poor appetite persisted. Computed tomography showed no improvement in the bilateral lower lobe lesions. Prednisone was initiated based on a clinical diagnosis of immunotherapy-related pneumonitis. The response to prednisone confirmed the diagnosis.Interventions:The patient first received ceftazidime and levofloxacin, but the symptoms persisted. Prednisone was initiated based on a clinical diagnosis of immunotherapy-related pneumonitis.Outcome:Complete resolution of the bilateral lung lesions occurred after 45 days of prednisone therapy.Conclusion:This case report highlights that both pneumonitis and bacterial pneumonia can occur as complications of anti-PD-1 immunotherapy.     

侵袭性肺曲霉菌病的临床诊治进展

侵袭性肺曲霉菌病是深部真菌感染性疾病，近年来发病率大大增加，死亡率极高，传统的诊断方法非常困难，治疗效果不佳。近年来，该病在诊断治疗上有很大的进展，本文就此进行综述。     

Possible relationship between organizing pneumonia and chronic pulmonary aspergillosis: A case report and literature review

Organizing pneumonia (OP) is a nonspecific response to various forms of lung injury and has been reported in association with several infectious agents. However, little is known about the relationship between OP and chronic pulmonary aspergillosis, and the mechanism of this linkage has not been elucidated. Here, we present a case of chronic pulmonary aspergillosis that led to the development of OP, which was successfully treated with corticosteroid and surgical intervention. In a review of the literature, we aim to highlight the possible relationship between OP and chronic pulmonary aspergillosis.     

戊型肝炎合并侵袭性曲霉菌病1例报告

1 病例介绍
　　患者男性,71岁,2010年9月10日以“右上腹不适,全身皮肤黄染伴尿色加深1个月余,发热2 d”为主诉入院。入院前1个月余患者无诱因出现右上腹不适、恶心、全身皮肤黄染、尿色加深,无寒战、高热,无腹胀、腹泻等。经常食用海鲜,2010年7月23日至本院查肝功能：ALT 2260 U ／L,AST 3328 U ／L, TBil 131．4μmol／L,DBil 103．9μmol／L;HEV －IgM阳性,HEV－IgG 阴性,抗－HBs 阳性,余病毒学指标均阴性;确诊为“急性黄疸型病毒性肝炎,戊型”,予保肝、降酶、退黄等治疗25 d后 ALT、AST 逐渐接近正常,但 TBil、DBil 进行性上升达410、333．5μmol／L。2010年8月19日开始予地塞米松10 mg 静脉滴注,1次／d,1周后复查肝功能示胆红素水平较前明显降低（TBil 256．3μmol／L,DBil 214．8μmol／L）,2010年8月26日将地塞米松减量至7．5 mg 静脉滴注,1次／d,治疗1周后复查肝功能示 TBil、DBil 较前稍有升高（TBil 308．2μmol／L,DBil 257μmol／L）,患者家属要求出院治疗。院外中医门诊就诊1周（期间不规律服用泼尼松片60～105 mg／d）后出现发热,最高体温39．0℃,伴咳嗽、黄白色痰,量多。中医门诊查肝功能ALT 157 U ／L,AST 128 U ／L,TBil 476μmol／L,DBil 365μmol／L;血常规：白细胞16．84×109／L,中性粒细胞百分比96．9％;为进一步诊治,2010年9月10日再次来本院,入院查体：神志清,精神差,全身皮肤、巩膜重度黄染,无肝掌,双肺可闻及湿罗音,右下肺明显。送痰涂片及培养、血培养等病原学检查,给予头孢曲松1 g,1次／12 h 抗感染;保肝、降酶、退黄等治疗。2010年9月11日血常规白细胞9．62×109／L,中性粒细胞百分比80．7％;ALT 165 U ／L,AST 141 U ／L,TBil 511．2μmol／L, DBil 360．5μmol／L;胸部 CT 示“两肺纹理粗乱,两中下肺可见斑片状阴影,右中下肺可见大片实变影,部分可见透光区”,细菌感染可能大,结合病史真菌感染也有可能,结核待排。头孢曲松治疗后患者体温正常2 d,第3天再次发热,最高体温38．8℃,咳嗽、咳灰黄色痰,量多,2010年9月13日将头孢曲松调整为亚胺培南1 g 静脉滴注,1次／12 h ＋莫西沙星400 mg 口服,1次／d,同时予两性霉素 B 雾化吸入预防真菌感染,但患者仍反复发热,最高体温38．9℃,咳嗽、咳痰较前无改善;复查白细胞13．17×109／L,中性粒细胞百分比90．8％;ALT 190 U ／L, AST 224 U ／L,TBil 295μmol／L,DBil 231μmol／L;2010年9月16日胸部 CT 示：两肺炎症较2010年9月11日有进展,左肺上叶及下叶背段实变伴空洞形成,考虑细菌、真菌重叠感染可能大。此时痰培养回示：肺炎克雷伯杆菌、铜绿假单胞菌、变形杆菌、曲霉菌;痰抗酸涂片阴性。遂予卡泊芬净首剂70 mg 后35 mg 静脉滴注,1次／12 h（Child 评分9分）＋伏立康唑首剂400 mg,静脉滴注,1次／12 h 后130 mg 静脉滴注,1次／12 h （患者体质量65 kg,Child 评分9分）抢先治疗。复查痰培养,4次均培养出曲霉菌。在卡泊芬净＋伏立康唑治疗第11天,亚胺培南＋莫西沙星治疗第14天时患者咳嗽、咳痰好转,但仍反复发热,最高体温38．5℃,且复查 CT 示肺部病灶消退不明显,可能是重叠有革兰阳性菌感染,再送痰培养,并于2010年9月27日开始加用利奈唑胺覆盖革兰阳性菌。加用利奈唑胺第3天,患者体温恢复正常,1周后复查胸部 CT 示两肺部炎症病灶较前有所吸收;痰涂片可见革兰阳性球菌、革兰阴性杆菌、真菌菌丝;痰培养仍示：肺炎克雷伯杆菌、铜绿假单胞菌、变形杆菌、曲霉菌,未培养出革兰阳性菌。后多次复查胸部 CT 示患者肺部多发炎症病灶较前逐渐吸收,限于患者经济原因,在卡泊芬净＋伏立康唑治疗47 d 后予以停用,调整为为伊曲康唑200 mg,静脉滴注,1次／d（首剂200 mg 静脉滴注,1次／12 h）使用16 d,后调整为伊曲康唑200 mg 口服,1次／12 h 治疗。治疗期间患者多次痰培养示：克雷伯杆菌、铜绿假单胞菌、变形杆菌;在亚胺培南＋莫西沙星使用2周后根据药敏予抗生素降阶梯调整为头孢哌酮舒巴坦＋阿米卡星,随患者临床症状改善,体温恢复正常,肺部病灶吸收,在头孢哌酮舒巴坦＋阿米卡星使用3周时调整为阿奇霉素口服。患者入院时激素用量为泼尼松片20 mg,3次／d,入院开始即予激素缓慢减量至停药,患者 TBil、DBil 逐渐恢复。2010年12月8日患者血常规白细胞5．37×109／L,中性粒细胞计数53．7％;ALT 11 U ／L,AST 31 U ／L,TBil 47．7μmol／L,DBil 32．9μmol／L,肺 CT 示炎症病灶较前明显吸收。患者要求出院,予以办理,嘱院外继续口服伊曲康唑抗真菌治疗（图1）。     

Primary vaginal malignant melanoma: A rare case report of successful treatment with nivolumab

Rationale:Primary vaginal malignant melanoma is a sporadic and very aggressive tumor that is treated through surgery or radiotherapy combined with chemotherapy. Since most cases are diagnosed at an advanced stage, the operation range is extensive, the quality of life is poor, and the prognosis is gloomy.Patient concerns:A 58-year-old woman presented irregular water-like leukorrhea for 1 month after 6 years of menopause. Positron emission tomography-computed tomography revealed a 3.1 × 2.6 × 3.2 mass on the middle and lower part of the right vaginal wall. A gynecological examination revealed a 2 to 3 cm exophytic black mass in the lower-right part of the vaginal orifice. This mass was 2 cm from the urethral orifice. Furthermore, the mucosa of the anterior inferior vaginal wall had blackened and thickened, and there were some scattered black dots at the medial labia minora.Diagnosis:Due to the patient''s symptoms with radiographic findings, the postmenopausal woman was diagnosed with primary vaginal malignant melanoma.Interventions:Surgery was done to remove the mass. The patient also underwent inguinal lymph node dissection, received immunotherapy, and was treated with nivolumab.Outcomes:After a 6-month follow-up period, the patient underwent a routine gynecological examination with negative radiological results. Moreover, no local recurrence or distant metastases were found.Lessons:This patient showed a good response to immunotherapy. With this treatment method, the prognosis is better for advanced-stage women, especially those who cannot endure the surgery. Local lesion resection and inguinal lymph node dissection combined with immunotherapy are recommended. The case reported here may help treat similar clinical cases.     

Prenatal diagnosis of pulmonary artery sling associated with tracheal agenesis: A case report

Pulmonary artery sling (PAS) and tracheal agenesis (TA) are rare diseases, and most cases of PAS are associated with tracheal bronchial malformations. However, PAS associated with TA is yet to be reported. We report a case of PAS with TA diagnosed prenatally. Due to the extremely low incidence, physicians do not have sufficient understanding of these diseases and it is challenging to diagnose these diseases by prenatal ultrasound, with high rates of misdiagnosis. Prenatal examination of the pulmonary artery branches, trachea, and esophagus is useful; therefore, improving the accuracy of prenatal diagnosis will help in perinatal management and counseling.     

常见变异型免疫缺陷病合并口普雷沃菌脓毒血症及肺曲霉菌感染1例报告

常见变异型免疫缺陷病(common variant immunodeficiency,CVID)是一种病因尚不十分清楚的原发性免疫缺陷病,由于其明显的异质性,临床上诊断较为困难。本文通过收集1例CVID患者诊治的临床资料,总结其临床特点和实验室结果,以进一步帮助临床医生提高对CVID的认识。     

Primary malignant melanoma of the esophagogastric junction: A case report

Rationale:Most gastrointestinal melanomas are metastatic from an oculocutaneous primary lesion; however, primary gastrointestinal melanomas have been found in all levels of the gastrointestinal tract. We present the case of Primary malignant melanoma of the esophagus and discuss the diagnostic methods, differentiation from metastatic lesions and treatment options.Patient concerns:A 78-year-old male patient presented with fresh blood vomiting and tarry stools for 1 day.Diagnoses:Esophagogastroduodenoscopy of this patient revealed a tumor ∼4 cm in size at the cardia side of the esophagogastric junction with dark-red and gray pigmentation. Immunohistochemical stains of the biopsy specimens were positive for S-100 and HMB-45, which are specific markers of melanoma.Interventions:Laparotomy with proximal gastrectomy was performed by the surgeon. Histological examination of the surgical specimen revealed the tumor arose from the distal esophagus with invasion of the proximal stomach. Primary malignant melanoma of the esophagus was diagnosed after a full skin and ophthalmic examination and positron emission tomography, which revealed no lesions elsewhere in the body.Outcomes:No tumor recurrence was noted at the 1-year follow-up.Lessons:Primary malignant melanoma of the esophagus is an extremely rare but highly aggressive tumor. The special pattern of pigmentation should be recognized while performing endoscopy. Early detection and radical resection of the tumor are critical to ensure favorable outcomes.     

Bilocular atrial malignant mesenchymoma causing mitral and localized pulmonary vein flow obstruction: diagnosis by transoesophageal echocardiography.

Cardiac malignant mesenchymoma is an extremely rare malignancy with poor prognosis. We report a patient presenting with a history and clinical findings typical of mitral stenosis. Transthoracic echocardiography showed a mass on the thickened posterior mitral leaflet. Transoesophageal echocardiography revealed two tumoural masses: one on the atrial side of the posterior mitral leaflet causing mitral obstruction, the other arising in the region of the right lower pulmonary vein orifice and obstructing inflow through this vein.     

Prenatal ultrasound diagnosis of fetal isolated right ventricular noncompaction with pulmonary artery sling: A rare case report

Noncompaction of the ventricular myocardium (NVM), also known as spongy myocardium, is a rare type of cardiomyopathy that has a serious impact on fetuses, children, and adults. NVM mainly affects the left ventricle, as isolated right ventricular noncompaction (IRVNC) is rare. Pulmonary artery sling (PAS) is a rare condition in which the left pulmonary artery anomalously originates from a normal positioned right pulmonary artery, and only a few studies have reported PAS in fetuses. Fetal IRVNC complicated with PAS has not been reported yet. Here, we report a case of IRVNC complicated with PAS that was diagnosed prenatally at 30 weeks gestation and confirmed by postpartum anatomy and pathology.     

A case report of malignant hyperthermia in a patient with myotonic dystrophy type I: A CARE-compliant article

Rationale:Several hereditary myopathies that can predispose to malignant hyperthermia (MH) are reported. However, the risk of MH in myotonic dystrophy type I (DM1) has been suggested equal to general population, although the evidence is limited to only a few case reports.Patient concerns:We encountered a rare case of MH during anesthesia induction with sevoflurane in a male adolescent with previously undiagnosed DM1.Diagnoses:After the event, genetic testing revealed the presence of a previously unknown heterozygous missense mutation in ryanodine receptor 1 (RYR1) associated with MH (c.6898T > C; p.ser2300Pro). Concomitantly, the patient was diagnosed with DM1 with abnormal cytosine-thymine-guanine triplet expansion in the DMPK gene.Interventions:Dantrolene was administered to treat the hypermetabolic manifestations in 20 minutes after the identification of MH.Outcomes:The patient was successfully treated and discharged without any complications. Laboratory abnormalities were recovered to baseline at postoperative 4 days.Lessons:The authors suggest that possible MH susceptibility in DM1 patients may be refocused. Genetic testing can be a screening tool for MH susceptibility in these population, prior to receiving general anesthesia.     

Successful treatment of pulmonary Nocardia farcinica infection with linezolid: case report and literature review

Nocardia infection is rare but potentially fatal. Therapy of Nocardia infection remains difficult. Linezolid, a novel oxazolidinone antibiotic, has proven to be effective, but clinical data are limited. Here we describe a case of a 45-year-old man with pulmonary N. farcinica infection following a liver transplantation. The initial therapy was trimethoprim-sulfamethoxazole, which showed no effect. According to susceptibility test, linezolid was administered with clearly improving the patient's condition. The treatment was stopped for anemia as drug related adverse event, and the therapy lasted for as long as 5 months. At the end of treatment clinical cure was confirmed and anemia reversed after discontinuation of linezolid. We also analyzed the clinical data of previously published reports by literature review, focusing on the efficacy and safety of linezolid treatment for Nocardia infection.     

Successful treatment of pulmonary infection due to Mycobacterium chelonae: case report and review.

During the past decade, Mycobacterium chelonae has been recognized with increasing frequency as a pulmonary pathogen. A review of previously reported cases reveals that most patients with pulmonary infections due to M. chelonae are nonimmunosuppressed but have underlying chronic lung disease. The infection is notably absent among blacks. M. chelonae organisms are characterized by a high degree of in vitro resistance to antituberculous drugs, and attempts at eradicating the organism through chemotherapy have been largely unsuccessful. The case of a 63-year-old previously healthy woman with progressive bilateral pulmonary disease due to M. chelonae is reported; she was treated successfully with a combination of cefoxitin and orally administered ciprofloxacin. Our experience supports the use of quinolones in combination with other active agents for the treatment of pulmonary infection due to M. chelonae.     

Isolated pulmonary amyloidosis: case report and review of the literature

The respiratory tract is rarely the site of localized tracheobronchial or nodular amyloid deposits. We present an unusual case of isolated nodular amyloidosis of the lung and review the current literature related to this disorder.     

Breast osteosarcoma originating from a malignant phyllodes tumor: A case report of a rare neoplasm

Rationale:Malignant phyllodes tumors with osteosarcomatous transformation are exceedingly rare. The clinical manifestations are similar to those associated with benign giant calcification, resulting in nonspecific and complex clinical symptoms.Patient concerns:A 59-year-old woman presented with a firm, painless, movable, 5.0 × 4.0 cm lump in the lower inner quadrant of the left breast that she had detected 1 month prior.Diagnoses:Breast osteosarcoma originating from a malignant phyllodes tumor was confirmed by histopathologic and immunohistochemical evaluation.Interventions:The patient underwent a wide local excision.Outcomes:The patient recovered uneventfully and was discharged after the operation. The 6-month postoperative follow-up assessment revealed no evidence of recurrence.Lessons:Diagnosing malignant phyllodes tumors with osteosarcomatous transformation requires a high level of suspicion and awareness by both surgeons and pathologists. They should be aware of the extent of such disease, which might be mistaken as benign giant calcification. Medical history and imaging findings are important for accurate diagnosis. Phyllodes tumor with an osteosarcomatous component is an aggressive neoplasm associated with distant metastasis. Delayed diagnosis and insufficient excision might negatively impact both treatment and survival.     

Successful treatment of pancreatic schwannoma by enucleation: A case report

Rationale:Pancreatic schwannomas are extremely rare and are difficult to diagnose preoperatively. Over the past 50 years, only 96 cases of pancreatic schwannoma have been reported in English literature. Herein, we report a case of pancreatic schwannoma treated with enucleation.Patient concerns:A 66-year-old woman visited a local hospital due to ventosities. Ultrasonography and computed tomography revealed a pancreatic mass. She visited our hospital for further diagnosis and treatment.Diagnosis and interventions:Magnetic resonance imaging revealed a tumor in the pancreatic body, and a solid pseudopapillary tumor was considered preoperatively. During the surgery, a pancreatic mass was found growing in the pancreatic body and tail. A successful tumor enucleation was performed. The mass was 7 × 6 × 3 cm in size with a thin capsule. Pathological examination revealed that the tumor was mainly composed of spindle-shaped cells with a palisading arrangement and no atypia. Both hypercellular and hypocellular areas were visible. Immunohistochemical staining showed that protein S-100 was strongly positive. The tumor was diagnosed as a benign schwannoma originating from the pancreatic body and tail.Outcomes:Postoperatively, the patient showed good recovery. During the 24-month follow-up period, the patient remained well and free of complications.Lessons:Pancreatic schwannomas are extremely rare and difficult to diagnose using imaging examinations. Enucleation is a safe and efficacious treatment for exophytic pancreatic schwannomas.