Received: 25 January 2022

Accepted: 21 June 2022

DOI: 10.1002/rcr2.1005

CASE REPORT

A case of cervical schwannoma with upper tracheal stenosis
followed up as asthma for 4 years
Akira Naomi1| Hideyuki Hujiwara2 | Tatsuhiko Koizumi2 | Yukari Sakurai2 |Yasuo Kohashi2 | Yukiko Yoneda2 | Yuka Kitamura1 | Yoshinobu Hattori1 |Yuji Saitou2
1
Department of Thoracic Surgery, Medical
Corporation Kiyosu Respiratory Medical Hospital,Haruhi Respiratory Medical Hospital, Achi, Japan2
Department of Respiratory Medicine, Medical
Corporation Kiyosu Respiratory Medical Hospital,Haruhi Respiratory Medical Hospital, Achi, JapanCorrespondence
Akira Naomi, Department of Thoracic Surgery,
Medical Corporation Kiyosu Respiratory MedicalHospital, Haruhi Respiratory Medical Hospital,
Haruhinagare 8-1, Kiyosu city, Aichi, Japan.
Email: akira.naomi@kiyosu-kokyuki.com

Abstract
Cases of upper tracheal stenosis due to cervical schwannoma are fairly rare; therefore,
no treatment has been determined. In this case, our patient had been treated forasthma for 4 years and was admitted to our hospital because of exacerbation. Computed tomography showed a tracheal stenosis lesion just below the vocal cords, and abiopsy revealed schwannoma. Conservative therapy was preferred rather than tumour
resection by surgery. Follow-up for 5 years showed no changes on imaging. Conservative treatment is considered as an option if the extratracheal tumour does not grow.
KEYWORDS
cervical schwannoma, conservative treatment, hot biopsy forceps, symptomatic asthma, tracheal stenosis

Associate Editor: Peter WarkINTRODUCTION
Tracheal stenosis due to cervical schwannoma is extremely
rare, and there is no established treatment method. Because
tumours are benign, there is no consensus as to whether
surgical resection or conservative treatment should beperformed. We report a case of cervical schwannoma withsevere upper tracheal stenosis.

CASE REPORTA 48-year-old woman was diagnosed with bronchial asthma
and had been followed up for 4 years. There were no special
notes on medical history or family history. At the time ofadmission, walking was possible with frequent breaks, andher level of dyspnea was considered to be IV degree modified British Medical Research Council (mMRC) according
to the Fletcher, Hugh–Jones classification. There was amildly palpable swollen tumour on the right side of the trachea and no other abnormal findings were found in blood,biochemical findings or tumour markers. Cervical computed tomography (CT) showed a solid tumour with amaximum diameter of 37 mm in the paratrachea which
excluded the surrounding tissue (Figure 1A). The tracheal
lumen was severely stenotic just below the cricoid cartilage(Figure 1B). A tumour biopsy was attempted with a bronchoscope, but no histopathology was obtained; therefore, apercutaneous paratracheal tumour biopsy was performed.
The tumour consisted of spindle-shaped cells, accompaniedby collagen fibres, and no mitotic figures or atypia wereobserved; immunostaining revealed CD34 ( ), S100 (+)
and β-catenin ( ), leading to the diagnosis of low-grade
schwannoma. Regarding the treatment policy, we first proposed surgery (partial thyroid cartilage resection plus firstto third rings of the trachea plus extra-cervical tumour
resection) to the patient, but she did not agree with it anddid not wish to have surgery. Therefore, as a symptomatictreatment, the portion of the tumour causing endoluminal
stenosis of the tracheal lumen was removed using hot
biopsy forceps under general anaesthesia. Approximately
1 ml of ethanol was injected into the residual intratracheal
tumour at five locations. Immediately after tumour removal
plus ethanol injection, the ventilator was withdrawn andher wheezing or respiratory distress (present before admission) improved. After about 1 month of rehabilitation, sheThis is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided theoriginal work is properly cited.
© 2022 The Authors. Respirology Case Reports published by John Wiley & Sons Australia, Ltd on behalf of The Asian Pacific Society of Respirology.
Respirology Case Reports. 2022;10:e01005.
https://doi.org/10.1002/rcr2.1005

wileyonlinelibrary.com/journal/rcr2

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NAOMI ET AL.

F I G U R E 1 (A) CT showed a solid tumour with a maximum diameter of 37mm in the paratrachea. (B) The tracheal lumen was severely stenotic. (C)The tumour with spindle shaped cells eventually was diagnosed as a low-grade schwannoma. (D) Intratracheal findings after second tumour removal plus
ethanol injection

F I G U R E 2 (A) Tumour regrowth in the bronchial lumen was observed 1 month later on relieving intratracheal stenosis. (B) No tendency for tumour
growth was observed on computed tomography 5 years later.

was discharged on foot. However, approximately 1 monthafter discharge (Figure 2A), tumour regrowth in the bronchial lumen was observed, and the tumour removal withhot biopsy forceps plus 1 ml of ethanol injection was performed. For the next 5 years, no tendency for tumour
growth was observed on CT (Figure 2B).

CERVICAL SCHWANNOMADISCUSSION
The number of cases of cervical schwannoma is relatively
small, and there are no reports of surgery for upper tracheal
stenosis due to cervical schwannoma. Therefore, it can be treated through multiple modalities such as primary tracheal resection or endoscopic treatment, cryotherapy, endoscopic
excision and microdebridement.1 Hamouri et al.2 advise thatthe choice of treatment should be made by the clinical presentation of the tumour (pedunculated vs. sessile), the risk oftracheal resection and the presence or absence of an extratracheal component. For this case, regarding the treatment ofcervical schwannoma, although we first proposed surgery, ourpatient desired symptomatic treatment mainly for the relief of
airway stenosis from the viewpoint of complications and cosmetology. Although rare, there are cases of malignant transformation from schwannoma,3–5 and if the paratracheal
tumour shows a rapid growth tendency, it is necessary to persuade the patient to reconsider surgery. Bronchoscopic ethanol
infusion therapy was first reported by Fujisawa et al.6 in 1986
for malignant tumours in the central airways. Ethanol injected
into the tumour (1) fixes the tumour tissue and shrinks thetumour itself, and (2) blocks the intravascular blood flow on
the surface of the tumour, leading to necrosis of the tumour.
Currently, it is used for malignant tumours such as haemangiomas and pancreas, cysts, lymphangioma, carcinoids and soon in addition to schwannoma,7–9 and in some cases, multipleinjections of ethanol are needed. The main complications havebeen reported to be drunkenness, pain, fever, transient hoarseness and cough,8,9 but no serious complications have beenreported and it is relatively simple and inexpensive. Therefore,
if the growth is gradual, ethanol injection may be considered
for extratracheal tumours, and symptomatic treatment may be
performed for intratracheal tumours by tumour removal plus
ethanol injection in cases where conservative treatment is considered an option.
A U T H O R C O N T R I BU T I O N
Akira Naomi is the primary author of the manuscript, anddrafted and revised the paper. Hideyuki Hujiwara, Tatsuhiko Koizumi, Yukari Sakurai, Yasuo Kohashi, YukikoYoneda, Yuka Kitamura, Yoshinobu Hattori and Yuji Saitou
are the attending doctors who treated the patient on admission. Yuka Kitamura diagnosed the patient pathologically.
All authors read and approved the final manuscript.
CONF LICT OF IN TEREST
None declared.

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DA T A A V A I L A B I L I T Y S T A T E M E N T
Data sharing is not applicable to this article as no new data
were created or analysed in this study.
ET H I C S S TA T E M E N T
The authors declare that appropriate written informed
consent was obtained for the publication of the manuscript
and accompanying images.
OR CI D
Akira Naomi

https://orcid.org/0000-0001-8185-905X

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How to cite this article: Naomi A, Hujiwara H,Koizumi T, Sakurai Y, Kohashi Y, Yoneda Y, et al. A
case of cervical schwannoma with upper tracheal
stenosis followed up as asthma for 4 years.
Respirology Case Reports. 2022;10:e01005. https://doi.
org/10.1002/rcr2.1005