Respirology Case Reorts

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April 2020
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Case Series
Clinical manifestations in patients with PI*MMMalton genotypes. A matter still unsolved in alpha-1 antitrypsin deficiency
Marina Aiello, Alberto Fantin, Chiara Longo, Ilaria Ferrarotti, Giuseppina Bertorelli, Alfredo Chetta
DOI: 10.1002/rcr2.528

The PI*MMMalton patients in our study showed a normal liver function, and none of them had renal function abnormalities or abdominal aortic aneurysm. Only a prevalence of lung disease has been detected.

Case Reports
Transnasal humidified rapid insufflation ventilatory exchange during bronchoscopy in severe pulmonary hypertension due to Gerbode defect
Lucas Upperman, Thomas Gildea, Ursula Galway
DOI: 10.1002/rcr2.519

Transnasal humidified rapid insufflation ventilatory exchange (THRIVE) has been shown to prevent desaturation and prolong apnoeic time in the perioperative setting but has not been used in patients with pulmonary hypertension due to concern for hypercapnia and resultant transient worsening of mean pulmonary artery pressure. Here, we describe a patient with severe pulmonary hypertension (mean pulmonary artery pressure of 82 mmHg) who underwent bronchoscopy using deep sedation with THRIVE with no recorded desaturation events and no post-operative signs of hypercapnia.

Severe acute respiratory failure due to a multifactorial diffuse alveolar haemorrhage
Ernesto Crisafulli, Barbara Burgazzi, Maria Majori, Walter Serra, Alfredo Chetta
DOI: 10.1002/rcr2.531

We here report the case of an 81-year-old male subject affected by a multifactorial diffuse alveolar haemorrhage (DAH), in which the bleeding was related to the administration of clopidogrel and warfarin, both implicated in the context of a polycythaemia. He developed a severe acute respiratory failure treated with a ventilatory support by means of a continuous positive airway pressure (C-PAP) therapy. An improvement of patient's clinical conditions was observed only after clopidogrel and warfarin discontinuation.

Unusual aetiology of lymphocyte-predominant exudative pleural effusion: primary mediastinal actinomycosis
Gil Myeong Seong, Chang Lim Hyun, Jee Won Chang, Changhwan Kim
DOI: 10.1002/rcr2.534

Mediastinal actinomycosis is a rare infectious condition and often mimics malignancy. A high level of clinical suspicion is needed to diagnose and cure actinomycosis in patients with indolent, unresolving, or relapsing chronic inflammatory disease.

Rare case of vascular ring with Kommerell diverticulum mimicking uncontrolled asthma
Ahmed Ben Saad, Nesrine Fahem, Houda Ben Saad, Asma Migaou, Chokri Kortas, Samah Joobeur
DOI: 10.1002/rcr2.535

We report a rare case of vascular ring with Kommerell diverticulum leading to a misdiagnosis of asthma in a 39-year-old woman.

Remarkable response to nivolumab in sarcomatoid malignant pleural mesothelioma with high PD-L1
Kazuya Tsubouchi, Shigesato Inoue, Ritsu Ibusuki, Takeshi Iwasaki, Taishi Harada
DOI: 10.1002/rcr2.536

We report herein a case of a 78-year-old male patient diagnosed with sarcomatoid malignant pleural mesothelioma (MPM) with high PD-L1 expression who was refractory to standard chemotherapy but had a remarkable and sustained response to nivolumab.

New perspectives for a huge traumatic pneumatocele treatment in a young adult
Sen-Ei Shai, Yi-Ling Lai, Hsiao-Wen Tang, Shih-Chieh Hung
DOI: 10.1002/rcr2.537

Traumatic pneumatocele (TP) is a very rare condition that occurs mostly in children and young adults. To the best of our knowledge, we present here a case of the largest TP without rib fracture. Under conservative treatment, the condition lasted for ~8 months before complete resolution. Detailed temporal changes in chest X-ray were recorded for the analysis of natural healing processes.

Silicone pneumonitis after gluteal filler: a case report and literature review
Boon Hau Ng, Wan Rahiza Wan Mat, Nik Nuratiqah Nik Abeed, Mohamed Faisal Abdul Hamid, Andrea Ban Yu-Lin, Chun Ian Soo
DOI: 10.1002/rcr2.538

We describe a case of severe silicone pneumonitis complicated with acute respiratory distress syndrome and bilateral pneumothorax secondary to silicon gluteal augmentation. In this case report, we aim to discuss our experience and approach in managing an uncommon case of silicone embolization syndrome.

Honeycomb lung-like structures resulting from clustering of traction bronchiectasis distally in sarcoidosis
Michiru Sawahata, Noriharu Shijubo, Takeshi Johkoh, Koichi Hagiwara, Satoshi Konno, Tetsuo Yamaguchi
DOI: 10.1002/rcr2.539

We herein describe the first case, to our knowledge, of pulmonary sarcoidosis with honeycomb lung-like structures developing as a result of concentration of traction bronchiectasis distally, predominantly in both lower lung fields, which developed through shrinkage of consolidations comprising a “central–peripheral band” detected in a woman in her 60s, with non-caseating epithelioid granuloma.

Pulmonary lymphangitic carcinomatosis from gallbladder cancer mimicking diffuse alveolar haemorrhage
Hisao Higo, Noriyuki Suzaki, Takuya Nagata, Taro Togami, Nobuya Ohara, Masaomi Marukawa
DOI: 10.1002/rcr2.540

An 81-year-old man was admitted due to dyspnoea and haemoptysis. We suspected diffuse alveolar haemorrhage based on computed tomography (CT) findings of diffuse bilateral ground-grass opacity and partial consolidation. Autopsy revealed pulmonary lymphangitic carcinomatosis of whole lungs and primary gallbladder cancer.

Severe clopidogrel-induced DRESS with eosinophilic pneumonia associated with Epstein–Barr virus reactivation
Yuji Inagaki, Kazunobu Tachibana, Yasushi Inoue, Takahiko Kasai, Yoshikazu Inoue
DOI: 10.1002/rcr2.541

We report a case of severe clopidogrel-induced drug reaction with eosinophilia and systemic symptom (DRESS) with concomitant eosinophilic pneumonia associated with reactivation of Epstein–Barr virus (EBV). The patient was treated with systemic corticosteroids and mechanical ventilation management. DRESS can progress to severe respiratory failure, and presence of EBV or herpes virus in the serum or resected lymph node tissue is an important finding for its diagnosis.

Durvalumab induced sarcoid-like pulmonary lymphadenopathy
Emma Sanderson, Hari Wimaleswaran, Clare Senko, Shane White, Christine F McDonald
DOI: 10.1002/rcr2.542

Immune checkpoint inhibitors, such as durvalumab, are associated with a variety of unique immune-related adverse events. In this case, sarcoid-like mediastinal and hilar lymphadenopathy developed with the use of adjuvant durvalumab for the treatment of lung adenocarcinoma.

Cholecystectomy under thoracic epidural anaesthesia for patient with severe intra-pulmonary shunt
Agustina Frankel, Rebekah Ferris, Peter Hodgkinson, Claire M Ellender
DOI: 10.1002/rcr2.543

Pulmonary arteriovenous malformations are common in patients with hereditary haemorrhagic telangiectasia and can be associated with significant hypoxia and intra-pulmonary shunt. We present a case of a young man with a known 57% calculated shunt requiring abdominal surgery and the multidisciplinary decisions required in the preoperative period to minimize post-operative complications.

Management of extensive subcutaneous emphysema using negative pressure wound therapy dressings
Nai-Chien Huan, Noorasyikin Mohamed Arifin, Teng-Shin Khoo, Yean-Chen Lai
DOI: 10.1002/rcr2.544

To introduce negative pressure wound therapy dressings (NPWTD) as a cheap, effective, safe, and novel technique in patients with extensive subcutaneous emphysema (SE), we report an 80-year-old gentleman who developed extensive SE post chest tube insertion for secondary spontaneous pneumothorax necessitating intubation. After exhaustion of other methods, he underwent NPWTD via two subcutaneous incisions made at chest wall. He responded well to NPWTD and was extubated three days later.

Vogt–Koyanagi–Harada disease during chemoimmunotherapy for non-small cell lung cancer
Yuri Kurono, Takayuki Takeda, Yusuke Kunimatsu, Nozomi Tani, Izumi Hashimoto, Kazuki Hirose
DOI: 10.1002/rcr2.545

Vogt–Koyanagi–Harada disease (VKHD) is very rare and drug-induced VKHD is quite rare. This would be the first report on VKHD during chemoimmunotherapy.

A case of primary pleural synovial sarcoma with endobronchial recurrence
Jen Lye Wan, Yoke Fong Lam, Kit Weng Foong, Norsalwa Abdul Ghani, Kumaresh Lachmanan
DOI: 10.1002/rcr2.547

Primary pleural synovial sarcoma (PPSS) is an extremely rare malignancy without a known cause. We report a case of a PPSS that was operated and recurred as an endobronchial metastasis.

Surgical treatment of micronodular thymoma with lymphoid stroma
Min Kyun Kang, Do Kyun Kang, Youn-Ho Hwang, Ji Yeon Kim
DOI: 10.1002/rcr2.548

Here, we describe the surgical treatment of extremely rare thymoma—micronodular thymoma with lymphoid stroma (MTWLS).

Clinical Image
Pneumonia in Parkinson's disease: barium aspiration in videofluoroscopic swallowing study
Kwang-Hwa Chang, Yu-Tien Tzeng, Jing-Hwa Wey, Yuan-Jun Liu, Yen-Nung Lin, Wen-Kuei Chung
DOI: 10.1002/rcr2.546

This is an excellent teaching image for daily clinical practice. Barium aspiration into tracheobronchial tree is a remarkable warning sign of dysphagia-related aspiration pneumonia. In patients with aspiration pneumonia, a thorough swallowing function assessment shall be considered. Also, along with clinical assessment, videofluoroscopic swallowing study is a good add-on tool for dysphagia treatment plan and shared decision-making for nasogastric tube placement to patients and their caregivers. Owing to atypical presentation in patients with Parkinson's disease, dysphagia is often overlooked. Therefore, extra attention should be paid.

Index

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2020 DecVol. 08 Issue 9
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