Presentation of Boerhaave’s syndrome as an upper-esophageal perforation associated with a right-sided pleural effusion: A case report

lNTRODUCTlON

Spontaneous esophageal rupture refers to the full-thickness rupture of the esophageal wall caused by indirect trauma, non-foreign bodies, non-esophageal, and/or adjacent organ disease; it is also known as Boerhaave’s syndrome[1]. While the incidence of this disease is low, it is easily misdiagnosed at an early stage and progresses rapidly. After the occurrence of an esophageal rupture, due to negative pressure in the pleural cavity, the stomach contents easily enter the mediastinum and thorax, which often causes serious mediastinum infection and empyema in the early stages. If not treated promptly, severe sepsis rapidly develops into multiple organ failure and even death, which is an emergency during thoracic surgery[2]. Therefore, the associated mortality rate is extremely high. Thus, correctly diagnosing spontaneous esophageal rupture in the early stage is of great importance for the survival of patients with spontaneous esophageal rupture.

Herein, we report a case of spontaneous esophageal rupture misdiagnosed earlier as pleural effusion at an early stage and investigated the causes of its misdiagnosis, along with our experience during diagnosis and treatment. We also highlight the importance of reviewing dynamic chest computed tomography (CT) scans for the diagnosis of spontaneous esophageal rupture.

His vital signs were stable and no other specific symptoms were noted.

CASE PRESENTATlON

Chief complaints

An 84-year-old male was admitted to a local hospital, with complaints of shortness of breath, abdominal distension, and vomiting.

1)建立模擬巷道精度驗證絕對坐標系。首先使用手持式激光測距儀反復測量驗證系統坐標系原點O，巷道中線在底面的投影作為驗證系統絕對坐標系的X軸，其正方向為掘進機試驗樣機的掘進方向，Y軸方向由精密測角儀測量X軸順時針偏轉90°方向，Z軸為O點豎直向上方向。

對稱矩陣在消元計算過程中有的一個很特別、但一直被忽視的特點：即規格化之前，第i行對角元以右元素與第i列對角元以下元素數值相等、位置對稱；規格化之后，第i行對角元以右元素與第i列對角元以下元素只相差一個對角元的比例系數，位置仍然對稱。因此在因子表的形成過程中，可僅計算對角元素和上三角元素，而下三角元素可按列通過規格化前的上三角元素賦值得到，從而省去大量下三角元素的計算及相應的除法計算，大大加快因子表的形成速度。

History of present illness

The patient was admitted to a local hospital, with complaints of shortness of breath, abdominal distension, and vomiting. He did not vomit again during his stay at the hospital.

ACEI類的藥物可降低血管緊張素Ⅱ濃度，因此最初用于治療高血壓，但在臨床長期應用過程中發現其對于腎臟具有明顯保護作用。在本次治療研究中，采取ACEI類藥物治療的IgA腎病患者的臨床總有效率達到90.5%以上，顯著高于非ACEI類藥物治療的對照組（71.5%），提示該種藥物治療可明顯保護腎功能，此外觀察組患者的血清清蛋白等水平均明顯優于對照組，提示該種藥物治療可降低甚至不利因子水平，進一步保護腎臟，研究結果與劉宏偉報道基本一致[2]。總之，對于IgA腎病治療，采取ACEI類藥物臨床療效顯著，保護患者腎功能。

After admission, the patient agreed to undergo right closed thoracic drainage and grapefruit-like residue drainage fluid was observed (Figure 1C). During the physical examination, subcutaneous emphysema of the right chest wall with crepitus was detected.

History of past illness

During surgery, a right lateral thoracic incision was made. The patient's right chest wall, muscles, and fascia were severely congested and edematous, along with a ruptured esophagus (Figure 1D).

Personal and family history

Spontaneous esophageal rupture, a rare and life-threatening disease, was first reported by Rokicki M in 1724, and to date, a mere 50 cases have been reported in the literature[3]. Based on an epidemiological survey for this disease in Iceland, it has a low incidence of 31 per million per year[4]. Moreover, several studies confirm that men are more prone to morbidity than women and that the highest risk group included those in the 40-60 years age group[5]. Spontaneous esophageal rupture caused by vomiting followed by a large meal often precipitates secondary bacterial infections, which contribute to 50% of the total mortality[6]. Therefore, early diagnosis and surgical treatment are important in the treatment of this disease.

Physical examination

(3)Glenn Selig was in Kabul on business related to his Florida public relations firm when he became one of at least 22 people killed during a 14-hour attack that began Saturday night and stretched into Sunday,his company reported in a statement.

Laboratory examinations

The initial routine blood examination results showed that both WBC count and CRP levels were slightly elevated (WBC: 12.15 x 10/L, CRP: 13.96 mg/L, Table 1).

During hospitalization, laboratory tests also indicated an increase in inflammatory markers.

Imaging examinations

The chest CT scan showed the presence of a small amount of fluid in the right pleural cavity(Figure 1A). Thus, chest CT scanning was repeated. Right-sided pleural effusion with right lung distension insufficiency and perforation of the upper left esophagus were observed (Figure 1B).

FlNAL DlAGNOSlS

Considering the above signs and symptoms, the patient was diagnosed with spontaneous perforation of the upper left esophagus, and an urgent esophageal hiatus repair was performed.

TREATMENT

The patient had no history of lung diseases.

OUTCOME AND FOLLOW-UP

Unfortunately, due to deterioration of his condition, the patient died from infection and respiratory failure.

Based on the evidence, the patient was diagnosed with pleural effusion and recommended to undergo right closed thoracic drainage; however, the patient's family refused given his advanced age.Therefore, antibiotics were prescribed to prevent infection. However, after treatment, blood inflammatory indicators were significantly elevated and did not improve [white blood cell (WBC) count: 19.40× 10/L; C-reactive protein (CRP): 304.90 mg/L] (Table 1). The patient also suffered from intermittent fevers and over time, complained of chest pain. After eight days, the patient was transferred to our hospital.

DlSCUSSlON

No similar disease was identified in his family.

總而言之，在新高考模式下，需要對傳統教學方式進行創新，以便提升教學質量。因此，在高中地理教學中，不僅要積極應用電子設備，注重引入國際性案例，提高學生眼界，而且還要根據具體教學內容，開展相應的實踐活動，進而可促使學生更靈活的應用地理知識，對其高考成績的提高具有重要意義。

The patient has provided informed consent for publication of the case. A copy of the written consent is available for review from the Editor-in-Chief of this journal.

To the best of our knowledge, spontaneous esophageal rupture often occurs in the thoracic esophagus and its incidence in the upper thoracic esophagus is relatively rare. The reasons for this are broadly described as follows: the myometrium of the esophagus is divided into two, the inner ring and the outer longitudinal layer. Approximately 2 mm thick elastic fibers are sandwiched between the two layers.Owing to the lack of coherence in the anatomical structure of the esophagus, a sudden rise in intraesophageal pressure (up to 290 mmHg) can lead to rupture at this altered anatomical structure of the esophagus[11]. While esophageal rupture occurs most commonly in the lower third of the left thoracic segment of the esophagus (80%), it is less frequent in the right esophagus, the upper thoracic, and ventral segments of the esophagus[12]. Among the physical signs, right pleural effusion is also uncommon. In the case of the upper thoracic esophageal perforation, prevertebral or subcutaneous air may be present[13]. Herein, we reported in detail, a case of a spontaneous esophageal rupture in the upper thoracic esophagus, with no obvious signs and symptoms in the early stage. Due to the lack of an early dynamic chest CT review, this disease was misdiagnosed.

Collectively, the reasons for the misdiagnosis were as follows: first, the on-admission chest CT report was only suggestive of a right-sided hydropneumothorax, inconsistent with CT presentation in most reports; additionally, chest pain began later during the course of disease progression, along with a lack of other typical manifestations. Finally, upper thoracic esophageal perforation is a rare site of esophageal rupture and the dynamic chest CT scan was not reviewed during hospitalization, thereby leading to early misdiagnosis and a consequent delay in appropriate treatment.

CONCLUSlON

This case report highlights the importance of a dynamic chest CT review, not only for initial identification of the injured segment but also for prioritizing subsequent treatment strategies. Moreover, we also provide clues for clinicians to recognize and diagnose spontaneous esophageal rupture at a rare site(upper-esophageal segment) by reporting this case of spontaneous esophageal rupture and summarizing the reasons for its misdiagnosis.

FOOTNOTES

All authors made substantial contributions to conception and design, acquisition of data, or analysis and interpretation of data; took part in drafting the article or revising it critically for important intellectual content; agreed to submit to the current journal; gave final approval of the version to be published; and agree to be accountable for all aspects of the work.

the Natural Science Foundation of Hubei Province, No. 2019CFB798.

Although many cases of spontaneous esophageal rupture have been reported, the lack of specific symptoms of this condition continues to pose a challenge[7]. Mackler's triad comprising an acute presentation of retching or vomiting, lower chest pain, and surgical emphysema, is a clinical manifestation with relatively high specificity for the diagnosis of spontaneous esophageal rupture.However, its incidence is only about 14%[8,9]. Other signs, which are non-specific, including hemodynamic blood instability or the presence of the Hammer sign-on auscultation, can also help in diagnosing the disease[10]. As secondary infection can irritate adjacent organs, symptoms including abdominal pain, nausea, chest tightness, shortness of breath, and dyspnea can also occur. In addition,elevated cardiac biomarkers and amylase also make it difficult to differentiate it from pericarditis,myocardial infarction, peptic ulcer, and other conditions. For patients with clinical suspicion of the disease, early chest CT examination is particularly important as it shows the manifestation of mediastinal or free peritoneal air as the first sign.

The authors have no conflicts of interest in relation to this work.

3.深推“兩學一做”，貫徹學習精神。公司領導班子以身作則、率先垂范，每月積極參加黨委中心組集中學習，各黨支部、黨小組通過講黨課、學習研討、知識競賽、黨員輪訓、撰寫學習心得等多種方式深入學習宣傳貫徹黨的十九大精神和習近平總書記系列重要講話精神及治國理政新理念新思想新戰略，不斷武裝頭腦、指導實踐，以高度的責任擔當精神，用實際行動和強力舉措推動黨的十九大精神落地生根、開花結果。

The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).

1.2 實驗設備 DWC150型動物實驗加壓艙，購于上海 701 所楊園醫用氧艙廠。MyLab 30CV型彩色超聲成像系統（LA435 超聲探頭），購于意大利百勝集團公司。BC-2800 Vet型全自動動物血液細胞分析儀，購于深圳邁瑞生物醫療電子股份有限公司。XL1000e 系列全自動凝血測試儀，購于北京眾馳偉業科技發展有限公司。

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溥儀沖齡登基，即使做了三載帝王，此時的他也不過是個五六歲的幼童。李凡的目光穿過人群，看著御座上的溥儀，只見他稚嫩而懵懂的眼神掃過身邊的太后、下面的臣子，對他們的悲傷憂愁難有感觸。

China

Ni Tan 0000-0002-7591-0143; Yin-Hua Luo 0000-0003-4381-7048; Guang-Cai Li 0000-0002-9728-0197;Yilin Chen 0000-0003-1969-9854; Wei Tan 0000-0001-7625-5518; Yue-Hua Xiang 0000-0002-9526-5778; Liang Ge 0000-0003-2147-7652; Di Yao 0000-0002-6966-0416; Ming-Hua Zhang 0000-0003-4577-626X.

Ma YJ

Webster JR

Ma YJ