INTRODUCTION: Dysphagia is a common presentation in older people. Diffuse idiopathic skeletal hyperostosis affecting the cervical spine is an uncommon cause of dysphagia and may be overlooked. CASE PRESENTATION: We present the case of an 88-year-old man with dysphagia and weight loss. Initial investigation with upper gastrointestinal endoscopy was inconclusive. A diagnosis of diffuse idiopathic skeletal hyperostosis as a cause for dysphagia was eventually made using video fluoroscopy. This showed a bony prominence impeding swallow at the level of C3. The patient was unfit for surgical management so a percutaneous endoscopic gastrostomy tube was inserted for feeding. CONCLUSION: The diagnosis of diffuse idiopathic skeletal hyperostosis involving the cervical spine often goes unrecognised as a cause of dysphagia despite its prevalence in the elderly population. Diagnosis is made using cervical radiographs, barium swallow and computed tomography. There is a risk of perforation with endoscopy in patients who have cervical diffuse idiopathic skeletal hyperostosis. Conservative management includes non-steroidal anti-inflammatory medications and a modified diet. Surgery may be considered in certain patients where conservative management fails

Iñaki Bovill

Natalia Ciapryna

Seema Srivastava

Journal of Medical Case Reports

English

PubMed

Abstract Introduction Dysphagia is a common presentation in older people. Diffuse idiopathic skeletal hyperostosis affecting the cervical spine is an uncommon cause of dysphagia and may be overlooked. Case presentation We present the case of an 88-year-old man with dysphagia and weight loss. Initial investigation with upper gastrointestinal endoscopy was inconclusive. A diagnosis of diffuse idiopathic skeletal hyperostosis as a cause for dysphagia was eventually made using video fluoroscopy. This showed a bony prominence impeding swallow at the level of C3. The patient was unfit for surgical management so a percutaneous endoscopic gastrostomy tube was inserted for feeding. Conclusion The diagnosis of diffuse idiopathic skeletal hyperostosis involving the cervical spine often goes unrecognised as a cause of dysphagia despite its prevalence in the elderly population. Diagnosis is made using cervical radiographs, barium swallow and computed tomography. There is a risk of perforation with endoscopy in patients who have cervical diffuse idiopathic skeletal hyperostosis. Conservative management includes non-steroidal anti-inflammatory medications and a modified diet. Surgery may be considered in certain patients where conservative management fails.</p

Srivastava Seema

Ciapryna Natalia

Bovill Iñaki

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Diffuse idiopathic skeletal hyperostosis as an overlooked cause of dysphagia: a case report

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CASE REPORT Open AccessCancerous pericarditis presenting as cardiactamponade in a 68-year-old man withpancreatic adenocarcinoma: a case reportSachie Kiryu1, Zensho Ito1*, Masashi Ishikawa1, Takafumi Akasu1,2, Yoshihiro Matsumoto1, Shinichi Hirooka2,Masayuki Saruta3 and Shigeo Koido1AbstractIntroduction: Pericardial effusion is a rare complication of pancreatic cancer. We report a case of cardiactamponade secondary to pancreatic cancer.Case presentation: A 68-year-old Japanese man was diagnosed as having pancreatic cancer during surgery andreceived chemotherapy for 28 months after the diagnosis. He was admitted to the emergency room with severedyspnea. Echocardiography revealed pericardial effusion with severe hypofunction. Emergency pericardial drainagewas performed to maintain hemodynamics, which resulted in the elimination of 450 mL of blood and themaintenance of circulatory dynamics. Cytological examination of the pericardial fluid revealed atypical cells andtumor cells suggesting adenocarcinoma.Conclusions: To our knowledge, pancreatic cancer complicated with cancerous pericarditis has not been previouslydocumented. This case highlights the extreme severity of pericardial effusion, a sign of progressive disease,secondary to pancreatic cancer. In the case of neoplastic pericardial effusion, an extremely poor prognosis must beconsidered.Keywords: Pancreatic cancer, Cardiac tamponadeIntroductionMalignant pericardial effusion caused by carcinomatouspericarditis is a complication of advanced malignancy.Pericardial effusion may cause cardiac tamponade andsudden death if not properly controlled. Cardiac tam-ponade as a complication of pancreatic cancer is veryrare. Cardiac tamponade caused by carcinomatous peri-carditis induces the retention of pericardial fluid, causingpericardial effusion, and must be treated promptly as anoncologic emergency [1]. Emergency treatment is neces-sary to prevent sudden death and relieve symptoms ofpericardial effusion in patients with neoplastic cardiactamponade [2]. To the best of our knowledge, pericardialinvolvement in patients with pancreatic cancer has neverbeen reported. Here, we report a case of metastatic pan-creatic cancer complicated by pericardial effusion.Case reportA 68-year-old Japanese man was suspected of havingpancreatic cancer 3 years previously due to an increasein carbohydrate antigen (CA)19-9. Abdominal computedtomography (CT) revealed a 26-mm hypoechoic masson the head of his pancreas and liver metastasis. Endo-scopic ultrasonography (EUS) and fine-needle aspiration(FNA) of the pancreatic mass revealed adenocarcinoma.He was diagnosed as having Stage IV unresectable© The Author(s). 2020 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License,which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you giveappropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate ifchanges were made. The images or other third party material in this article are included in the article's Creative Commonslicence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commonslicence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtainpermission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to thedata made available in this article, unless otherwise stated in a credit line to the data.* Correspondence: zensho@jikei.ac.jp1Division of Gastroenterology and Hepatology, Department of InternalMedicine, The Jikei University School of Medicine, Kashiwa Hospital, 163-1Kashiwa-shita, Kashiwa, Chiba 277-8567, JapanFull list of author information is available at the end of the articleKiryu et al. Journal of Medical Case Reports          (2020) 14:213 https://doi.org/10.1186/s13256-020-02546-ypancreatic cancer, and gemcitabine (GEM) plus nano-particle albumin-bound paclitaxel (nab-PTX) doubletchemotherapy was administered. After 2 years of GEM/nab-PTX chemotherapy, progression of the disease wasconfirmed; therefore, the chemotherapy regimen waschanged to GEM + tegafur-gimeracil-oteracil (S1). Sixmonths later, he experienced dyspnea and visited theclinic after a few days of experiencing symptoms. CT re-vealed pericardial effusion and bilateral pleural effusion.Laboratory tests revealed elevated C-reactive protein(CRP) at 6.23 mg/dL. Pneumonia was treated with beta-lactam antibiotics. Therefore, he was diagnosed ashaving pneumonia and heart failure. The following day,his blood oxygen saturation was 90% on room air, andhe had tachycardia (110 beats per minute) and hyper-thermia (37.2 °C). His blood pressure was 86/67 mmHg.Respiratory and circulatory failure was managed in anemergency room. Electrocardiography revealed sinustachycardia, and echocardiography revealed a severepericardial swinging heart motion (Fig. 1a and b). Peri-cardial drainage was performed through a pericardialwindow, resulting in the drainage of 450 mL of blood,and the pericardial fluid was subjected to cytologicalexamination. Cytological examination of the fluid re-vealed tumor cells indicating adenocarcinoma. Bloodlevels of CA19-9 and carcinoembryonic antigen (CEA)were 5.8 ng/mL and 50 IU/mL, respectively, which grad-ually increased during the course of treatment, suggest-ing pancreatic cancer progression. A pericardial drainagetube was removed after improvement in his general con-dition. One week after the surgical procedure, intraven-ously administered chemotherapy with GEM/nab-PTXwas re-initiated. Sixteen days after re-initiation ofchemotherapy, he was admitted to the emergency roomand died of cardiac arrest due to severe respiratory fail-ure without evidence of recurrent pericardial effusion.In this case, a pathological autopsy was performed. Onmacroscopic examination, the parietal pericardium andvisceral pericardium had adhered, indicating cancerouspericarditis (Fig. 2a). Pericardial effusion was not ob-served. On microscopic examination, a large number oftumor cells had infiltrated the pericardium, and somehad infiltrated the lymph vessels of the myocardium(Fig. 2b and c). These results suggest pericardial metas-tasis of pancreatic cancer.DiscussionCarcinomatous pericarditis is observed in 10–20% of pa-tients with malignancies, with the most common malig-nancies being lung cancer, breast cancer, leukemia, andmalignant lymphoma. According to a study by Bergeand Sievers, malignant tumor metastasis involving thepericardium was observed in 50% of autopsies of pa-tients with metastatic malignant tumors [3]. Pericardialmetastasis does not frequently cause cardiac tamponade.However, cardiac tamponade is a life-threatening condi-tion requiring prompt diagnosis and treatment. Cardiactamponade is the lethal phase of neoplastic pericarditisresulting from cardiac compression and the accumula-tion of pericardial fluid. Intrapericardial pressure in-creases and cardiac relaxation is inhibited, leading to lowoutput; patients often experience dyspnea, hypotension,tachycardia, cold sweats, and fatigue [4]. When symp-toms occur, echocardiography should be performedsince it is the most useful examination for diagnosingpericardial effusion and detecting signs of tamponade.Although the diagnosis of cardiac tamponade is basedon established clinical criteria, an accurate and earlydiagnosis of tamponade can often be established usingechocardiography [5].When neoplastic cardiac tamponade is diagnosed,drainage by pericardiocentesis should be performed torelieve symptoms and prevent sudden death. A high rateof recurrence (40–70%) has been reported due to thepersistence of carcinomatous pericarditis-induced peri-cardial effusion [6]. Therefore, systemic and localizedFig. 1 a Computed tomography findings. Extensive pericardial effusion and bilateral pleural effusion. b Echocardiography (apical four-chamberview) findings. An echo-free space around the heart revealed severe pericardial effusion. LA left atrium, LV left ventricle, RA right atrium, RVright ventricleKiryu et al. Journal of Medical Case Reports          (2020) 14:213 Page 2 of 4treatment should be provided to suppress pericardialeffusion. Localized treatment includes percutaneousdrainage, anticancer agent injection to destroy pericar-dial adhesions, and surgical resection of the pericardium.The most appropriate procedure should be selectedbased on the patient’s general condition. Intrapericardialtreatment should be tailored to the type of tumor. Forexample, tris(1-aziridinyl)phosphine sulfide (Thiotepa) isrecommended for patients with breast cancer, and cis-platin is recommended for patients with lung cancer [7].The course after pericardial drainage has been reportedto vary according to the type of cancer [4].Malignant pericardial effusion is associated with anunfavorable prognosis. Wang et al. and Gross et al. re-ported that the mean survival times after detection ofmalignant pericardial effusion by pericardiocentesis were3.1 and 3.7 months, respectively [8, 9]. To the best ofour knowledge, no reports of intrapericardial treatmentor endocardial treatment in patients with pancreatic can-cer are available. We continued only systemic treatmentbecause intrapericardial treatment could have causeddiastolic dysfunction.Several mechanisms regarding primary tumor metasta-sis to the pericardial cavity have been proposed, includ-ing lymphatic spread, hematogenous spread, systemicspread via nerves, and direct extension. Most tumorsmetastasize to the heart through the mediastinal lymphnodes. An autopsy was performed in this case, and amain lesion in the head of the pancreas with extensivefibrosis was observed. Moreover, active tumor cellsremained. Vessel invasion and nerve invasion were ob-served, which were considered to have caused the metas-tasis to other organs. In the heart, the pericardium andmyocardium showed strong tumor adhesion, which wasconsidered indicative of cancerous pericarditis. Tumorcell infiltration was observed around almost the entirepericardium, and extensive lymphatic and nerve invasionwas observed. Tumor cells also spread to the lymphaticvessels in the heart parenchyma, and some had infil-trated between myocardial fibers. Reports indicate thataminoglycoside antibacterial agents reduced cardiac con-tractility [10]. In this case, these types of antibiotics werenot used, and the occurrence of cardiac dysfunction wasconsidered unlikely. From the pathological results, theinfiltration of cancer cells into the myocardium was con-sidered remarkable, and the progression of the cancercaused a decrease in cardiac function, ultimately leadingto death. Similar to that in the pericardium, severelymphatic invasion and venous invasion were observedin several organs. Therefore, the tumor was consideredto have reached the pericardium via a lymphatic orhematogenous route where it formed an infiltrating le-sion. In the lung, metastasis formation, intravasculartumor embolism, and pleural dissemination wereFig. 2 a Cancerous pericarditis: the parietal pericardium and visceral pericardium adhered to each other (arrow heads) and exhibited tumorinfiltration (arrow). b Hematoxylin and eosin-stained pericardial tissue. A large number of tumor cells infiltrated the pericardium. Tumor cellsinfiltrated the lymph vessels of the myocardium (× 20). c Hematoxylin and eosin-stained pericardial tissue. A large number of tumor cellsinfiltrated the myocardium (× 20). d Immunostained (D2–40) pericardial tissue. Findings of pericardial lymphatic invasion and lymphaticmetastasis (× 100)Kiryu et al. Journal of Medical Case Reports          (2020) 14:213 Page 3 of 4observed. The bronchiolar and bronchial walls showedhigh degrees of tumor invasion, and the peripheral air-way was constricted, likely causing respiratory failure.Debilitation occurred throughout the body due to cach-exia. Hepatic dysfunction occurred due to tumor infiltra-tion into the liver. Thus, in this case, the cause of deathwas considered tumor metastasis, including pericardialmetastasis, throughout the body. Pericardial metastasisof pancreatic cancer may suggest cancer metastasisthroughout the whole body.ConclusionPericardial metastasis and cardiac tamponade in pancre-atic cancer are extremely rare. In this case, lymphaticand vascular invasion caused metastasis to the pericar-dium and myocardium. In our patient, the interval be-tween tamponade and death was very short (42 days).Pericardial metastasis may be an indication of systemicmetastases, and tamponade can be considered an indica-tor of a poor prognosis. These possibilities are importantto consider when pericardial effusion develops in pa-tients with pancreatic cancer.Authors’ contributionsSKi and ZI wrote the manuscript. MI and TA analyzed and interpreted thepatient’s images, managed the case, and edited the manuscript. SHperformed the histological examination and was a major contributor towriting the manuscript. SKi and SKo clinically treated the patient. SKo andMS critically reviewed the manuscript and provided significant assistance inwriting the manuscript. All authors have read and approved the final versionof the manuscript.FundingThe authors did not obtain specific grant funding for this research. Theauthors declare that they have no competing interests.Ethical approval and consent to participateThis study was conducted in accordance with the basic principles of theDeclaration of Helsinki.Consent for publicationWritten informed consent was obtained from the patient’s next of kin forpublication of this case report and any accompanying images. A copy of thewritten consent is available for review by the Editor-in-Chief of this journal.Competing interestsNone declared.Author details1Division of Gastroenterology and Hepatology, Department of InternalMedicine, The Jikei University School of Medicine, Kashiwa Hospital, 163-1Kashiwa-shita, Kashiwa, Chiba 277-8567, Japan. 2Department of Pathology,The Jikei University School of Medicine, Kashiwa Hospital, Kashiwa City, Chiba277-8567, Japan. 3Division of Gastroenterology and Hepatology, Departmentof Internal Medicine, The Jikei University School of Medicine, Tokyo 105-8461,Japan.Received: 14 August 2020 Accepted: 2 October 2020References1. Vaitkus PT, Herrmann HC, LeWinter MM. Treatment of malignant pericardialeffusion. JAMA. 1994;272:59–64.2. Buck M, Ingle JN, Giuliani ER, Gordon JR, Therneau TM. Pericardial effusionin women with breast cancer. Cancer. 1987;60:263–9.3. Berge T, Sievers J. Myocardial metastases. A pathological andelectrocardiographic study. Br. Heart J. 1968;30:383–90.4. Takayama T, Okura Y, Okada Y, Honma K, Nashimoto A, Sato N, et al.Characteristics of neoplastic cardiac tamponade and prognosis afterpericardiocentesis: a single-center study of 113 consecutive cancer patients.Int J Clin Oncol. 2015;20:872–7.5. Cheitlin MD, Armstrong WF, Aurigemma GP, Beller GA, Bierman FZ, Davis JL,et al. ACC/AHA/ASE 2003 guideline update for the clinical application ofechocardiography--summary article: a report of the American College ofCardiology/American Heart Association Task Force on Practice Guidelines(ACC/AHA/ASE Committee to Update the 1997 Guidelines for the ClinicalApplication of Echocardiography). J Am Coll Cardiol. 2003;42:954–70.6. Adler Y, Charron P, Imazio M, Badano L, Baron-Esquivias G, Bogaert J, et al.2015 ESC Guidelines for the diagnosis and management of pericardialdiseases. Kardiol Pol. 2015;73:1028–91.7. Adler Y, Charron P, Imazio M, Badano L, Baron-Esquivias G, Bogaert J, et al.2015 ESC Guidelines for the diagnosis and management of pericardialdiseases: The Task Force for the Diagnosis and Management of PericardialDiseases of the European Society of Cardiology (ESC) Endorsed by: TheEuropean Association for Cardio-Thoracic Surgery (EACTS). Eur Heart J. 2015;36:2921–64.8. Wang ML, Liao WB, Bullard MJ, Lin FC, Lin PJ, Chiang CW, et al. Cardiactamponade in Taiwan. Jpn Circ J. 1997;61:767–71.9. Gross JL, Younes RN, Deheinzelin D, Diniz AL, Silva RA, Haddad FJ. Surgicalmanagement of symptomatic pericardial effusion in patients with solidmalignancies. Ann Surg Oncol. 2006;13:1732–8.10. Adams HR et al. Cardiac toxicities of antibiotics. Environ Health Perspect.1978;26:217-23.Publisher’s NoteSpringer Nature remains neutral with regard to jurisdictional claims inpublished maps and institutional affiliations.Kiryu et al. Journal of Medical Case Reports          (2020) 14:213 Page 4 of 4

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Diffuse idiopathic skeletal hyperostosis as an overlooked cause of dysphagia: a case report

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