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\begin{document}

\title{Patient With Spondyloarthropathy Presenting with Subglottic Stenosis: A
Case Report}



\author[1]{Akanksha  sharma}%
\author[2]{Aman  Goyal}%
\author[3]{Navroop Momi}%
\author[1]{julius Birnbaum}%
\affil[1]{UPMC Mercy}%
\affil[2]{Seth GS Medical College and KEM Hospital}%
\affil[3]{Christian Medical College and Hospital Ludhiana}%


\vspace{-1em}



  
  \date{June 16, 2024}


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\textbf{TITLE}

Patient With Spondyloarthropathy Presenting with Subglottic Stenosis: A
Case Report

\textbf{KEY CLINICAL MESSAGE}

Our case highlights the occurrence, management, and progression of
subglottic stenosis in a patient with spondyloarthropathy. Dual
management strategy, involving medical therapy and endoscopic
intervention has yielded satisfactory outcomes.

\textbf{INTRODUCTION}

Subglottic stenosis (SGS) is a component of laryngotracheal stenosis
that involves the narrowing of the airway below the vocal cords.
Symptoms of SGS include wheezing, dyspnea, hoarseness, stridor, and, in
severe cases, asphyxiation {[}1,2{]}. It may be caused by congenital,
acquired, or iatrogenic factors, with acquired cases often attributed to
infection, trauma, or idiopathic causes {[}3{]}. In patients with
laryngotracheal stenosis, the most common cause is iatrogenic factors,
accounting for 54.7\% of cases, with 59\% developing subglottic stenosis
{[}4{]}. Autoimmune diseases are one of the less common causes of SGS
and include granulomatosis polyangiitis, relapsing polychondritis,
inflammatory bowel disorders, rheumatoid arthritis, sarcoidosis, and
amyloidosis. A diagnosis of idiopathic SGS is made when there is no
identifiable underlying systemic disease, and it is typically a
diagnosis of exclusion. Despite the availability of effective endoscopic
and open surgical interventions, the inflammatory aspect of the
condition continues to hinder many patients from achieving complete
remission {[}2{]}. A detailed medical history is essential for patients
with respiratory issues because misidentifying the condition can lead to
treatment delays and life-threatening consequences.

Our study aimed to report an atypical subglottic stenosis associated
with spondyloarthropathy. While this condition is commonly associated
with various etiologies, such as trauma, iatrogenic factors, or
idiopathic causes, its occurrence in the context of spondyloarthropathy
remains relatively less understood. This case report sheds light on the
importance of early recognition and management to improve the quality of
life of affected individuals.

\textbf{CASE HISTORY}

The patient was diagnosed with subglottic stenosis in 2017. Her chief
complaint was difficulty breathing. Pulmonary function tests showed an
obstructive pattern of FEV1/FVC at 73\%; DLCO was also reduced to 70\%,
thought to be due to anatomic emphysema, but no imaging findings of
emphysema.

Her medical and surgical history was significant for osteoarthritis (
Celebrex), hyperlipidemia, degenerative disc disease (cervical, lumbar),
hypertension, meningioma, hypothyroidism, cholecystectomy, appendectomy,
knee replacement, finger surgery, tonsillectomy, and mastectomy.

\textbf{INVESTIGATIONS AND TREATMENT}

Figure 1 demonstrates the pre-dilatation subglottic view of the patient.
The patient underwent balloon dilatation, CO2 excision, Kenalog
injection, and mitomycin administration in January 2017. Figure 2
demonstrates the immediate post-dilatation subglottic view. These
interventions led to a 70\% improvement in breathing.

She underwent similar treatment a year later, in July 2018, followed by
October 2019.

In December 2020, she presented with swelling, pain, and morning
stiffness around the bilateral 2nd-4th MCPs. Considering the son's
suspected peripheral spondyloarthropathy and inflammatory bowel disease,
the underlying autoimmune processes were questionable, and she was
referred to the rheumatology clinic for further workup. The patient
tested negative for ANCA and MGUS. There had no history of saddle nose,
asthma, or inflammatory bowel disease.

On workup, she tested positive for ANA Hep 2, and a PET scan showed
diffuse uptake (including dens), including the shoulders, knees, and 1st
MTP supportive of spondyloarthropathy. She had been receiving 15 mg of
methotrexate (MTX) since September 2021, hoping it would alter the
course of her subglottic stenosis, but this did not help. Despite
starting MTX therapy in September 2021, she required dilatation in the
early summer of 2022. Leflunomide was added in July 2022.

As of January 2023, the last dilation was performed ten months ago. She
intermittently experienced dyspnea. In addition, no joint pain was
observed. Upon reviewing her laboratory results, we noticed a gradual
decrease in white blood cell count (WBC count=3100
/mm\textsuperscript{3}) after starting leflunomide 20 mg in July 2022.
In May 2023, it was discontinued owing to increased leukopenia.
Furthermore, we escalated therapy with TNF alpha inhibitors and
initiated cimzia therapy.

She had undergone six dilatations and steroid injections.

Her last dilation procedure was performed in February 2022. However, the
patient noted that her overall dyspnea improved significantly.

\textbf{Figures:}

\textbf{Figure 1: Pre dilatation subglottic view}

\textbf{Figure 2: Post dilatation subglottic view}

\textbf{CONCLUSION AND RESULT}

Our case highlights the occurrence, effective management, and natural
progression of subglottic stenosis in a patient with
spondyloarthropathy. Given her son's history of inflammatory bowel
disease (IBD) and peripheral spondyloarthropathy, screening for IBD with
colonoscopy to rule out occult illnesses is crucial. With above
mentioned interventions, patients overall dysonoea had improved
significantly.

\textbf{DISCUSSION}

Subglottic stenosis is diagnosed after careful clinical and laboratory
examination. The respiratory symptoms are nonspecific and present late
in disease progression, necessitating a high clinical suspicion level
{[}5{]}. In a retrospective study by Dion et al., out of 32 patients, 3
had autoimmune subglottic stenosis due to RA as the etiology. Other
autoimmune etiologies were systemic lupus erythematosus and
granulomatosis with polyangiitis, to name a few {[}6{]}. Consistent with
our findings, subglottic stenosis appears to be associated with an
underlying autoimmune etiology.

Treatment modalities have evolved from open surgical procedures to
endoscopic dilation, laser surgery, and adjuvant intralesional steroid
injections {[}7,8{]}. Interval dilation is the mainstay of treatment for
idiopathic subglottic stenosis. In a study conducted by Menapace et al.,
they concluded that open surgery should only be reserved for refractory
cases of subglottic stenosis, which had postoperative adverse effects
such as a change in voice quality {[}9{]}. Recurrence was common in
their study, highlighting the need for continued long-term follow-up in
such patients, and our patient was scheduled for semi-annual follow-up.
Generally, treatments are tailored to the severity and recurrence of a
patient's breathing and, possibly, voice dysfunction. Patients with
autoimmune subglottic stenosis and severe intraluminal narrowing may
require tracheostomy as a treatment modality {[}10{]}. In our patient,
neither open surgery nor tracheostomy was needed, and was managed with a
combination of endoscopic and medical therapy. There are controversial
opinions on the use of immunotherapy for managing subglottic stenosis,
and methotrexate has shown a prolonged interval before the requirement
of another interval dilation session in patients {[}11{]}. Leflunomides
can also be used for this purpose {[}11{]}.

According to some reports, IBD may also be related to subglottic
stenosis, which is also linked to lupus erythematosus, amyloidosis,
Wegener granulomatosis, and tuberculosis {[}11{]}. Considering the
suspected peripheral spondyloarthropathy and inflammatory bowel disease
in the patient, there were questionable underlying autoimmune processes.
The ESSG and Amor criteria, which were created for the complete SpA
group, including peripheral SpA, were compared with ASAS criteria, which
were devised for individuals with SpA with prominent peripheral signs
after the development of the ASAS criteria for axial SpA {[}12{]}.

The patient was initially diagnosed as having idiopathic subglottic
stenosis. However, later assessments revealed peripheral joint
involvement of the shoulders, knees, and first metatarsophalangeal
joint, and a PET scan demonstrated diffuse uptake (including dens).
These findings supported the diagnosis of spondyloarthropathy, and the
patient was diagnosed with a subglottic stenosis accompanied by an
underlying systemic disorder. For this variant, TNF-\selectlanguage{greek}α \selectlanguage{english}inhibitors were
efficacious, and the patient was prescribed and responded well.

Our case highlights the occurrence, effective management, and natural
progression of subglottic stenosis in a patient with
spondyloarthropathy. Given her son's history of inflammatory bowel
disease (IBD) and peripheral spondyloarthropathy, screening for IBD
using colonoscopy to rule out occult illnesses is crucial.

Owing to the risk of sudden airway compromise possible with subglottic
stenosis, our case highlights the importance of physicians being aware
of this rare complication for quick diagnosis and effective management.
However, a dual management strategy, involving medical therapy and
endoscopic intervention, is generally sufficient. Long-term follow-up
should be scheduled with the patient to reassess their status
periodically.

\textbf{Author contricbutions:}

AS- Writing and editing of the manuscript, AG- Writing, reviewing and
editing, NKM - Writing, reviewing and editing, KP- Writing, reviewing
and editing, JB - Supervision, writing, reviewing and ediitng.

\textbf{Funding Statement} : The authors have no funding to disclose.

\textbf{Ethical Compliance} : Ethical approval was not applicable for
this study design

\textbf{Data Access Statement} : No datasets were used for this
manuscript

\textbf{Conflict of Interest Declaration} : The authors declare that
they have no affiliations with or involvement in any organization or
entity with any financial interest in the subject matter or materials
discussed in this manuscript.

\textbf{CONSENT}

Written informed consent was obtained from the patient to publish this
report in accordance with the journal's patient consent policy.

\textbf{References}

1. Pasick LJ, Anis MM, Rosow DE. An Updated Review of Subglottic
Stenosis: Etiology, Evaluation, and Management. Curr Pulmonol Rep.
2022;11(2):29-38. doi: 10.1007/s13665-022-00286-6. Epub 2022 Mar 3.
PMID: 35261874; PMCID: PMC8892813. 2. Rosow DE, Barbarite E. Review of
adult laryngotracheal stenosis: pathogenesis, management, and outcomes.
Curr Opin Otolaryngol Head Neck Surg. 2016 Dec;24(6):489-493. doi:
10.1097/MOO.0000000000000305. PMID: 27585080. 3. Jagpal N, Sommerfeldt
J, Shabbir N. Subglottic Stenosis. 2023 Jul 26. In: StatPearls
{[}Internet{]}. Treasure Island (FL): StatPearls Publishing; 2023 Jan--.
PMID: 33085412. 4. Gelbard A, Francis DO, Sandulache VC, Simmons JC,
Donovan DT, Ongkasuwan J. Causes and consequences of adult
laryngotracheal stenosis. Laryngoscope. 2015 May;125(5):1137-43. doi:
10.1002/lary.24956. Epub 2014 Oct 7. PMID: 25290987; PMCID: PMC4562418.
5. Aravena C, Almeida FA, Mukhopadhyay S, et al. Idiopathic subglottic
stenosis: a review. J Thorac Dis. 2020;12(3):1100-1111.
doi:10.21037/jtd.2019.11.43 6. Dion GR, De la Chapa JS, Bettenhausen W,
Dominguez LM, Simpson CB. Differing Progression to Posterior Glottic
Stenosis in Autoimmune and Idiopathic Subglottic Stenosis. The
Laryngoscope.;131(8): 20211816-1820. doi:10.1002/lary.29085 7. Feinstein
AJ, Goel A, Raghavan G, et al. Endoscopic Management of Subglottic
Stenosis. JAMA Otolaryngology--Head \& Neck Surgery.
2017;143(5):500-505. doi:10.1001/jamaoto.2016.4131 8. Comparative
Treatment Outcomes for Patients With Idiopathic Subglottic Stenosis
\textbar{} Laryngology \textbar{} JAMA Otolaryngology--Head \& Neck
Surgery \textbar{} JAMA Network. Accessed September 11, 2023. 9.
Menapace DC, Modest MC, Ekbom DC, Moore EJ, Edell ES, Kasperbauer JL.
Idiopathic Subglottic Stenosis: Long-Term Outcomes of Open Surgical
Techniques. Otolaryngol Head Neck Surg. 2017;156(5):906-911.
doi:10.1177/0194599817691955 10. Causes and consequences of adult
laryngotracheal stenosis - Gelbard - 2015 - The Laryngoscope - Wiley
Online Library. Accessed September 11, 2023. \emph{com}11. Hoffmann RM,
Kruis W. Rare Extraintestinal Manifestations of Inflammatory Bowel
Disease. Inflammatory Bowel Diseases. 2004;10(2):140-147.
doi:10.1097/00054725-200403000-00013. 12. Akgul O, Ozgocmen S.
Classification criteria for spondyloarthropathies. World J Orthop.
2011;2(12):107-115. doi:10.5312/wjo.v2.i12.07\selectlanguage{english}
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