Respirology Case Reorts

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April 2024
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Case reports
Successful embolization of a clinically significant pulmonary arteriovenous malformation
Pedro Fernandes, João P Silva, Pedro Patrão, António Reis, Teresa Belo
DOI: 10.1002/rcr2.1330

We present the clinical case of a 59-year-old female patient with dyspnoea and a history of peripheral desaturation, which was detected in the perioperative period 4 years earlier. Although she didn't present any typical symptoms such as platypnea, epistaxis, telangiectasias or hemoptysis, the chest-CT scan was compatible with the diagnosis of pulmonary arteriovenous malformation. An angiography confirmed the diagnosis and a selective embolization of the afferent artery was performed with resolution of symptoms.

Unilateral pulmonary agenesis with ipsilateral facial congenital anomalies in an adult with obstructive airway disease
Nilanthi Gunathilaka, Harshana Bandara, Tharindi Dissanayaka, Sathiaruban Sathiyamoorthy, Wathsala Gunasinghe, Saman Kularatne
DOI: 10.1002/rcr2.1332

Lung agenesis is a rare entity to diagnose in adult pulmonology. Here we present an adult who presented with asthma with unilateral lung agenesis along with ipsilateral facial deformity.

A case of primary pleural epithelioid hemangioendothelioma achieving stable disease with paclitaxel treatment: A case report and literature review
Chun-Ying Chou, Hsiang-Wei Hu, Tom Wei-Wu Chen, Shu-Yung Lin
DOI: 10.1002/rcr2.1341

Pleural epithelioid hemangioendothelioma (EHE) is exceptionally rare, and the prognosis for pleural EHE is grim, typically resulting in a survival of less than 1 year. In this case report, we present the case of a 45-year-old woman who endured 1 month of right-sided pleuritic chest pain, was diagnosed with right pleural EHE, and achieved an 11-month progression-free survival following paclitaxel treatment.

The utility of endobronchial ultrasound-guided transbronchial mediastinal cryobiopsy (EBUS-TBMC) for the diagnosis of mediastinal lymphoma
Nai-Chien Huan, Wei Lun Lee, Hema Yamini Ramarmuty, Larry Ellee Nyanti, Melody Shu Ling Tsen, Dahziela Yunus
DOI: 10.1002/rcr2.1342

Endobronchial ultrasound-guided transbronchial mediastinal cryobiopsy (EBUS-TBMC) is a novel technique that can provide larger samples with preserved tissue architecture, with an acceptable safety profile. We present a gentleman with anterior mediastinal mass who underwent EBUS-transbronchial needle aspirate (TBNA) followed by EBUS-TBMC. TBMC samples provided adequate tissue to attain a diagnosis of primary mediastinal large B-cell lymphoma.

Endobronchial cryptococcosis without pulmonary parenchymal involvement caused by Cryptococcus neoformans in an immunocompetent host
Yui Shimanuki, Susumu Sakamoto, Hiromichi Yoshida, Noboru Iizuka, Shion Miyoshi, Satoshi Sonobe, Naobumi Tochigi, Kazuma Kishi
DOI: 10.1002/rcr2.1343

In this article, we report a case of endobronchial cryptococcosis caused by C. neoformans without pulmonary parenchymal involvement in an immunocompetent host.

Tuberous sclerosis complex associated lymphangioleiomyomatosis caused by de novo mutation of TSC2 gene in Vietnam: A case report
Dinh Van Luong, Le Ngoc Huy, Nguyen Xuan Giang, Nguyen Huu Hong Thu, Nguyen Hai Ha, Nguyen Huy Binh
DOI: 10.1002/rcr2.1346

Herein, we describe an early 20s female admitted to the Hospital with dyspnea, chest pain, hypopigmented macules, and facial fibroadenomas. She has a medical history of renal angiomyolipomas (ALM) and pneumothoraces. Diagnosis with LAM was confirmed through high-resolution computed tomography (HRCT) scan and histopathology of lung biopsy. Whole exome sequencing analysis identified a frameshift mutation c.4504del (p.L1502Cfs*74) in the patient's TSC2 gene. This variant was de novo due to its absence in the patient's parents. This is the first report on the clinical and genetic etiology of TSC-LAM in Vietnam.

Misdiagnosis of lymphoma as vasculitis: A case report
Shushan Wei, Haobin Hu, Haoyue Helena Lan, Na Li, Qingling Zhang
DOI: 10.1002/rcr2.1347

This article describes a patient with NKTCL misdiagnosed as vasculitis who presented with sinusitis, abdominal pain, anorexia, and lung shadows, and also presented with eosinophilia, which was misdiagnosed as eosinophilic granuloma. The article describes the clinical features, diagnostic methods and differential diagnosis of lymphoma and highlights the importance of a multidisciplinary approach in accurate diagnosis and treatment.

Pulmonary artery pseudoaneurysm after thoracic radiation therapy: A case report and review of the literature
Gerdinique (G C) Maessen, Thijs (T W) Hoffman, Lidwien (L) Graat-Verboom, Marc (M) van Leersum, Hans-Jurgen (J J) Mager
DOI: 10.1002/rcr2.1349

A pulmonary pseudo-aneurysm can present with moderate to massive haemoptysis. Potential causes include trauma, infection, cancer, or iatrogenic causes such as (chemo)radiotherapy. The gold standard for diagnosis is a CTA of the chest with contrast in pulmonary circulation, and endovascular embolization is an effective treatment in most cases.

Recurrence of sarcoidosis accompanied by lung cancer after drug-induced pulmonary sarcoidosis with lung injury
Kentaro Suzuki, Yoshiro Kai, Ryosuke Kataoka, Maiko Takeda, Shigeo Muro
DOI: 10.1002/rcr2.1351

We present the case of sarcoidosis recurrence associated with lung cancer after drug-induced pulmonary sarcoidosis with lung injury. To our knowledge, this is the first report of sarcoidosis triggered by drug administration and lung cancer. Histological diagnosis of mediastinal lymphadenopathy with lung cancer is essential for differentiating metastasis from sarcoidosis.

Acute myocardial infarction with e-cigarette or vaping-use associated lung injury in a young Filipino vape user
Margarita Isabel C Fernandez, Margaret Francine Co, Janine Bianca Marie Rafael, Regiel Christian Mag-usara, Vanessa Ediza, Rafael Luis Gavino, Jan Christian Feliciano, Nigel Jeronimo Santos, Mark Andrian Yano, Julian Alexander Huibonhoa, Richard Henry Tiongco, Lenora Fernandez
DOI: 10.1002/rcr2.1353

This is the case of a young Filipino male with no known comorbidities and a two-year history of daily vape use, who presented with acute myocardial infarction from 2 vessel coronary artery disease. The patient also developed e-cigarette or vaping-use associated lung injury (EVALI) manifesting as respiratory distress and consolidation pneumonia with negative microbial detection studies. This case highlights a possible association between vape use and the development of both acute lung injury and myocardial infarction.

Effective treatment of steroid-resistant immune checkpoint inhibitor pneumonitis with mycophenolate mofetil
Nao Shioiri, Ryota Kikuchi, Itsuka Matsumoto, Kanako Furukawa, Kenichi Kobayashi, Shinji Abe
DOI: 10.1002/rcr2.1356

Insufficient evidence is available for treating steroid-resistant immune checkpoint inhibitor pneumonitis (CIP). Although guidelines recommend the use of immunosuppressants, the efficacy of mycophenolate mofetil (MMF) has not been sufficiently verified. Herein, we report two cases of steroid-resistant CIP that improved with the co-administration of mycophenolate mofetil (MMF).

A challenging case of drug-related acute fibrinous and organizing pneumonia: A rare case report
Chenxia Guo, Wei Wu, Xiang Zhu, Qingtao Zhou, Ying Liang
DOI: 10.1002/rcr2.1357

Acute fibrinous and organizing pneumonia (AFOP) is a rare type of interstitial pneumonia. We present a case of AFOP probably induced by anti-prostate cancer medications.

Achieving complete metabolic response in stage IV lung adenocarcinoma with chemotherapy, nivolumab, ipilimumab, and salvage SBRT: A case report
Salvador Gamez Casado, Javier David Benitez Fuentes, Beatriz Álvarez Rodríguez, Gema García Ledo
DOI: 10.1002/rcr2.1362

A 55-year-old male with stage IV oncogene-negative, PDL1-negative lung adenocarcinoma demonstrated a complete metabolic response to a novel treatment strategy. This regimen combined double immune checkpoint inhibition (ICI) with chemotherapy, followed by stereotactic body radiotherapy (SBRT), after initial partial response to two cycles of carboplatin, pemetrexed, nivolumab, and ipilimumab, plus maintenance with nivolumab and ipilimumab. This case underscores the effectiveness of integrating multiple treatment modalities for non-small cell lung cancer (NSCLC).

Clinical images
The case of huge simple pulmonary aspergilloma that responded remarkably to drug therapy
Makoto Yokota, Junji Takiguchi, Hiromi Tomioka
DOI: 10.1002/rcr2.1337

We report a case of simple pulmonary aspergilloma accompanied by a large tomour shadow that improved with pharmacological treatment alone.

Multiple pulmonary nodules with diffuse idiopathic pulmonary neuroendocrine cell hyperplasia and minute pulmonary meningothelial-like nodules
Tsuyoshi Sasada, Ryo Tachikawa, Shigeo Hara, Keisuke Tomii
DOI: 10.1002/rcr2.1344

We report the first case report of concurrent diffuse idiopathic pulmonary neuroendocrine cell hyperplasia (DIPNECH) and multiple pulmonary meningothelial-like nodules (MPMNs). A 78-year-old woman presented with multiple pulmonary nodules, mixed with solid and ground-glass nodules. Pathology confirmed that the multiple pulmonary nodules were a combination of (DIPNECH) and (MPMNs).

Ruptured giant thoracic hydatid cyst presenting with respiratory symptoms
Reza Ershadi, Hesam Amini, Ghazal Roostaei, Hossein Kazemizadeh, Leila Mollamohammadi, Niloofar Khoshnam Rad
DOI: 10.1002/rcr2.1345

We present the case of a 22-year-old college student who presented with progressive dyspnea, chest pain, fever, cough, and hemoptysis. Imaging revealed a giant cystic lesion and “water lily” sign within the left hemithorax. A giant hydatid cyst was removed by posterolateral thoracotomy. Thorough history, physical examination, and appropriate imaging studies, are crucial for accurate diagnosis.

Diffuse alveolar haemorrhage due to atypical hemolytic uremic syndrome (aHUS) associated with COVID-19
Atif Siddiqui, Amanda Tchakarov
DOI: 10.1002/rcr2.1350

We present a fatal case of delayed presentation of atypical hemolytic uremic syndrome (aHUS) in a 78-year-old female post-COVID-19. Delayed presentation of atypical HUS after COVID-19 with diffuse alveolar haemorrhage is uncommon and can be life threatening.

Efficacy of dupilumab as an alternative to corticosteroids in the treatment of exacerbations of allergic bronchopulmonary aspergillosis
Masamitsu Hamakawa, Tadashi Ishida
DOI: 10.1002/rcr2.1354

The pathogenesis of ABPA involves not only eosinophils but also plasma cells that produce IgE, which is responsible for type I allergic mechanisms. Dupilumab may be an effective alternative to corticosteroids because it inhibits T cell to plasma cell differentiation by blocking IL4.

Respiratory maze: An anatomical variant of tracheal bronchus
Dedeepya Gullapalli, Shivendra Tangutoori, Subramanya Shyam Ganti
DOI: 10.1002/rcr2.1355

Tracheal bronchus is a rare congenital abnormality of bronchial variation arising from trachea. Special attention should be paid during intubation as it can potentially obstruct the bronchus.

Successful ultrathin bronchoscopy with cryobiopsy for diagnosing and removing mucus plugs in allergic bronchopulmonary mycosis mimicking lung cancer
Yuki Takigawa, Hiromi Watanabe, Ken Sato, Mayu Goda, Tomoyoshi Inoue, Miho Fujiwara, Suzuka Matsuoka, Kenichiro Kudo, Akiko Sato, Keiichi Fujiwara, Takuo Shibayama
DOI: 10.1002/rcr2.1359

We present a case of successful ultrathin bronchoscopy with cryobiopsy for diagnosing and removing mucus plugs in a patient with allergic bronchopulmonary mycosis mimicking lung cancer.

Arthrogryposis multiplex congenita causing hypercapnic respiratory failure and airway obstruction in an adult patient
Harshana Bandara, Sathiaruban Sathiyamoorthy, Dinusha Withana, Tharindi Dissanayake, Harshani Athapaththu, Wathsala Gunasinghe
DOI: 10.1002/rcr2.1361

This case highlights arthrogryposis multiplex congenita (AMC) as a rare cause of hypercapnoeic respiratory failure and airway obstruction in adults and emphasizes the usage of leak-port in tracheostomy-NIV (non-invasive-ventilation) specially in resource poor setting when the tracheostomy-NIV mask is unavailable.

Index

2024 AprVol. 12 Issue 4
2024 MarVol. 12 Issue 3
2024 FebVol. 12 Issue 2
2024 JanVol. 12 Issue 1
2023 DecVol. 11 Issue 12
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