| Case Series |
A family history of Hermansky–Pudlak syndrome complicated with pulmonary fibrosis: a case series and review
Katiuska Liendo Martinez, Fernando Pedraza, Marta Fuentes Alonso, Luis Puente Maestu, Carla Rodriguez Naranjo, Javier De Miguel-Diez DOI: 10.1002/rcr2.720
The Hernansky–Pudlak syndrome (HPS) is a rare genetic disorder with autosomal recessive inheritance characterized by oculocutaneous albinism and bleeding diathesis, and rarely lung involvement. We report three cases diagnosed with HPS from the same family of 12 siblings, with six albinos, of whom four and the father had pulmonary fibrosis. We believe these are interesting cases to report, especially for the lack of cases registered in Spain. |
| Case Reports |
Post-mortem biopsy of a patient with late exacerbation of COVID-19 pneumonia
Kuniko Takahashi, Koichiro Kajiura, Michitaka Nasu, Kei Nakamura, Kazuki Sugata, Akiko Matsuzaki DOI: 10.1002/rcr2.724
Here, we describe pathological findings detected through post-mortem percutaneous needle biopsy of a patient with late-onset severe pneumonia from coronavirus disease (COVID-19). |
The diagnostic challenge of pneumocystis pneumonia and COVID-19 co-infection in HIV
Alistair G B Broadhurst, Usha Lalla, Jantjie J Taljaard, Elizabeth H Louw, Coenraad F N Koegelenberg, Brian W Allwood DOI: 10.1002/rcr2.725
We present the case of a 54-year-old male with newly diagnosed advanced HIV who presented with clinical, biochemical, and radiological features that were consistent with both pneumocystis pneumonia (PCP) and coronavirus disease 2019 (COVID-19). He subsequently tested positive for both diseases; however, the contribution of each disease to his progressive respiratory failure is unclear. |
Video-assisted thoracic surgery for a case of chronic progressive pulmonary aspergillosis undergoing haemodialysis complicated by anorexia nervosa: a case report
Toru Shirahata, Maho Akimoto, Kentaro Minegishi, Shunsuke Endo, Hidetoshi Nakamura, Makoto Nagata DOI: 10.1002/rcr2.727
In this report, we present a rare case of a Japanese woman with anorexia nervosa who developed pulmonary aspergillosis, and describe the clinical and pathophysiological characteristics of pulmonary aspergillosis in the patient. We also made comparisons with previous reports. |
Successful bronchial arterial infusion chemotherapy combined with radiotherapy for an endobronchial metastasis after resection of small cell lung cancer
Yoshitsugu Horio, Yozo Sato, Hiroyuki Tachibana, Waki Hosoda, Junichi Shimizu, Toyoaki Hida DOI: 10.1002/rcr2.728
We report a case of an endobronchial polypoid metastasis after resection of small cell lung cancer (SCLC). |
Complete response in a patient with lung cancer suffering from three pembrolizumab-induced immune-related adverse events including retinal vasculitis
Chihiro Mimura, Motoko Tachihara, Sentaro Kusuhara, Hidenori Fukuoka, Yoshihiro Nishimura DOI: 10.1002/rcr2.730
We report a case of non-small cell lung cancer with a long-term response associated with multiple immune-related adverse events caused by pembrolizumab, such as retinal vasculitis, hypophysitis, and thyroiditis. |
Congenital bronchobiliary fistula: a case report and literature review
Le Thuong Vu, Nguyen Minh Duc, Thieu-Thi Tra My, Mai Tan Lien Bang, Le Tuan Linh, Tran Quynh Huong DOI: 10.1002/rcr2.731
Congenital bronchobiliary fistula (CBBF) is a rare, anomalous fistula that occurs between the respiratory and biliary systems, with unknown aetiology. Here, we present the epidemiology and clinical features of a CBBF case in a two-day-old baby girl that was initially diagnosed as non-resolving pneumonia. Computed tomography (CT) and fistulography under the guidance of bronchoscopy were used to obtain the correct diagnosis. Surgical excision of the fistulous tract was performed, with complete recovery. |
New pneumothorax complicating successful treatment of persistent air leak by endobronchial valves
Yiu Cheong Yeung, Yu Hong Chan, Man Ying Ho, Ming Chiu Chan, Hau Chung Kwok, Wai Cho Yu DOI: 10.1002/rcr2.732
The present case report included two cases of pneumothorax after endobronchial valve treatment of persistent air leak (PAL) complicating pneumothorax. The occurrence of this adverse event (the first case showed new ipsilateral pneumothorax and the second case showed new contralateral pneumothorax) shed light to the underlying mechanism of this complication and has not been reported before in the treatment of PAL. |
Multimodality treatment in immunocompromised patients with severe COVID-19: the role of IL-6 inhibitor, intravenous immunoglobulin, and haemoperfusion
Nophol Leelayuwatanakul, Napplika Kongpolprom, Thitiwat Sriprasart, Vorakamol Phoophiboon, Vorawut Thanthitaweewat, Sarita Thawanaphong, Worawan Sirichana, Naricha Chirakalwasan, Kamon Kawkitinarong, Chanchai Sittipunt, Opass Putcharoen, Leilani Paitoonpong, Gompol Suwanpimolkul, Watsamon Jantarabenjakul, Nattachai Srisawat, Monvasi Pachinburavan DOI: 10.1002/rcr2.733
We report two cases of immunocompromised patients with severe coronavirus disease 2019 (COVID-19)-related cytokine release syndrome requiring mechanical ventilation who were treated with multimodality treatment consisting of tocilizumab, intravenous immunoglobulin (IVIG), and haemoperfusion. Within 48 h, both patients showed clinical improvement with PaO2:FiO2 ratio and haemodynamic stability. Both survived to discharge. There were no adverse events following these therapies. |
Multiple infected lung bullae associated with Mycobacterium intracellulare in a Japanese man
Tatsuya Kodama, Atsushi Kurokawa, Hiroyuki Kokuto DOI: 10.1002/rcr2.734
Here, we report the first documented case of multiple infected lung bullae associated with Mycobacterium avium complex (MAC) in a 29-year-old man. |
Snowball-like appearance on radial endobronchial ultrasonography in a patient with invasive mucinous adenocarcinoma
Takeshi Kawaguchi, Daisuke Himeji, Kiichio Beppu, Kousuke Marutsuka DOI: 10.1002/rcr2.735
Invasive mucinous adenocarcinoma (IMA), which is a relatively rare lung adenocarcinoma, is difficult to diagnose by computed tomography. No reports of radial endobronchial ultrasonography (EBUS) findings in IMA have been published. This article presents a case of IMA with a characteristic shadow, snowball-like appearance on radial EBUS in a 67-year-old Japanese man. |
| Clinical Image |
Natural closing of a tracheal tear caused by intubation in a patient with relapsing polychondritis
Hiroshi Handa, Shinya Azagami, Masamichi Mineshita DOI: 10.1002/rcr2.721
Airway involvement is an important predictive factor for relapsing polychondritis (RP). RP often develops into severe tracheobronchial stenosis with malacia. Although tracheal tears rarely occur, we report a tracheal tear post intubation, which healed without surgery. |
Diffuse alveolar haemorrhage due to pheochromocytoma crisis
Naoki Shijubou, Toshiyuki Sumi, Koki Kamada, Yuichi Yamada, Hisashi Nakata, Hirofumi Chiba DOI: 10.1002/rcr2.722
Alveolar haemorrhage due to pheochromocytoma should be considered as a diagnosis for interstitial shadows with extreme hypertension. |
Chest wall fistula in a patient with pyothorax-associated lymphoma
Saeko Takahashi, Kenshiro Omura, Morio Nakamura DOI: 10.1002/rcr2.723
Fistula formation is an uncommon but serious therapeutic complication of pyothorax-associated lymphoma (PAL) because it decreases the quality of life in patients. Further, a collapsed lung may predispose to pneumonia. In PAL, the lesions might invade the skin and optimal irradiation dose, region, and timing should be carefully determined. |
Endobronchial vasculitis in childhood granulomatosis with polyangiitis
Matthew D Wong, Leanne M Gauld DOI: 10.1002/rcr2.729
Laryngo-tracheo-bronchial disease in childhood granulomatosis with polyangiitis may acutely present with endobronchial small vessel vasculitis without airway stenosis. Treatment should not be delayed in the presence of haemoptysis as it may indicate acute pulmonary capillaritis which can lead to fatal respiratory failure from diffuse alveolar haemorrhage. |
Novel pulmonary perfusion imaging using chest digital dynamic radiography for pulmonary artery sarcoma
Shota Yamamoto, Fumio Sakamaki, Genki Takahashi, Ryotaro Yuji, Tomohiro Matsumoto, Terumitsu Hasebe DOI: 10.1002/rcr2.737
Chest digital dynamic radiography (DDR) is a novel method for evaluating pulmonary perfusion and ventilation. It could depict ventilation–perfusion mismatch in a pulmonary artery sarcoma with severe stenosis in the right pulmonary artery. This report is the first demonstration of ventilation–perfusion mismatch in a malignant neoplasm using DDR. |
Abnormal anastomosis of peripheral pulmonary arteries, initially suspected to be an arteriovenous fistula, in an adult with Williams syndrome
Hironori Oyamatsu, Kenichi Watanabe, Ryota Kiriyama, Takehiko Okagawa, Seijirou Niimi DOI: 10.1002/rcr2.738
Here, we present the case of a 45-year-old man with Williams syndrome with suspected right pulmonary arteriovenous fistula, wherein A10b forming the loop appeared to run near A6 and flow into the upper pulmonary vein. Angiography findings revealed that the loop was an abnormal anastomosis from A6c to A10b. Peripheral pulmonary artery stenosis is present in 40% of the patients with Williams syndrome. In this case, basal pulmonary artery was narrowed. This abnormal anastomosis might have been a bypass from A6c to A10b that formed as a compensation for poor blood flow in the basal lung. |
Complete lung collapse as a rare complication of sarcoidosis-associated mediastinal lymphadenopathy
Abdul R Rishi, Adel Zurob, Christopher Williams, Hussam Al-Sharif, Ali Zaied, Vipul Trivedi, Muhammad A Rishi DOI: 10.1002/rcr2.739
Here, we report an extremely rare case of a 47-year-old African American female with complete lung collapse associated with sarcoidosis. Although lymphoma should be ruled out when patients with mediastinal lymphadenopathy develop lung collapse, sarcoidosis should be considered in the differential, especially when associated with fibrosing mediastinitis. |
