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A Case of Tracheal Stenosis As an Isolated Form of Immunoproliferative Hyper-Igg4 Disease in a 17-Year-Old Girl

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A Case of Tracheal Stenosis As an Isolated Form of Immunoproliferative Hyper-Igg4 Disease in a 17-Year-Old Girl children Case Report A Case of Tracheal Stenosis as an Isolated Form of Immunoproliferative Hyper-IgG4 Disease in a 17-Year-Old Girl Natalia Gabrovska 1,*, Svetlana Velizarova 1, Albena Spasova 1, Dimitar Kostadinov 1, Nikolay Yanev 1, Hristo Shivachev 2, Edmond Rangelov 2, Yanko Pahnev 2, Zdravka Antonova 2, Nikola Kartulev 2, Ivan Terziev 3 and Kaloyan Gabrovski 4 1 Department of Pulmonary Diseases, Multiprofile Hospital for Active Treatment of Pulmonary Diseases “St. Sofia”, Medical University–Sofia, 1431 Sofia, Bulgaria; [email protected] (S.V.); [email protected] (A.S.); [email protected] (D.K.); [email protected] (N.Y.) 2 Department of Pediatric Thoracic Surgery, Pediatric Surgery Clinic, University Multiprofile Hospital for Active Treatment and Emergency Medicine “N. I. Pirogov”, 1606 Sofia, Bulgaria; [email protected] (H.S.); [email protected] (E.R.); [email protected] (Y.P.); [email protected] (Z.A.); [email protected] (N.K.) 3 Department of Pathology, University Hospital ‘’Tsaritsa Uoanna–ISUL”, 1527 Sofia, Bulgaria; [email protected] 4 Department of Neurosurgery, University Hospital “St. Ivan Rilski”, Medical University–Sofia, 1431 Sofia, Bulgaria; [email protected] * Correspondence: [email protected]; Tel.: +359-887-931-009 Abstract: Immunoglobulin G4-related disease (IgG4-RD) is a lymphoproliferative disease which is described almost exclusively in adults. There are only a few pediatric patients who have been observed with this disorder. Here, we describe a rare case of IgG4-RD in a 17-year-old girl with Citation: Gabrovska, N.; Velizarova, a single manifestation—tracheal stenosis without previous intubation or other inciting event. She S.; Spasova, A.; Kostadinov, D.; Yanev, had mixed dyspnea and noisy and weakened breathing. Immunoproliferative hyper-IgG4 disease N.; Shivachev, H.; Rangelov, E.; Pahnev, Y.; Antonova, Z.; Kartulev, was diagnosed, based on elevated serum IgG4 and histological findings. Until now we have chosen N.; et al. A Case of Tracheal Stenosis to treat the girl only with corticosteroids with a good response so far. The general condition as as an Isolated Form of well as the respiratory function are regularly monitored. The tracheal involvement of IgG4-RD is Immunoproliferative Hyper-IgG4 uncommon. Nonetheless, it is a manifestation that should be included in the differential diagnosis of Disease in a 17-Year-Old Girl. Children tracheal stenosis. 2021, 8, 589. https://doi.org/ 10.3390/children8070589 Keywords: tracheal stenosis; children; IgG4 immunoproliferative disease Academic Editor: Joaquim M. B. Pinheiro Received: 29 May 2021 1. Introduction Accepted: 9 July 2021 Published: 12 July 2021 Immunoglobulin G4-related disease (IgG4-RD) is a lymphoproliferative disorder which is observed almost exclusively in adults. There are very few pediatric patients who have been described with the disease [1,2]. Publisher’s Note: MDPI stays neutral with regard to jurisdictional claims in It is an immune-mediated condition that can affect almost any organ in the body. published maps and institutional affil- Common presentations are major salivary and lacrimal gland enlargement, orbital disease, iations. autoimmune pancreatitis (the first described involved organ) and tubulointerstitial nephri- tis [3,4]. It is characterized by a lymphoplasmacytic infiltrate composed of IgG4+ plasma cells, exuberant fibrosis and mild to moderate eosinophilia [5,6]. Usually, the most common cause of acquired tracheal stenosis is a previous intubation, tracheotomy or tracheostomy. Here, we describe a rare case of IgG4-RD in an adolescent Copyright: © 2021 by the authors. girl with a single manifestation—tracheal stenosis without previous intubation or other Licensee MDPI, Basel, Switzerland. This article is an open access article inciting event. distributed under the terms and 2. Case Presentation conditions of the Creative Commons Attribution (CC BY) license (https:// We present a case of 17-year-old girl diagnosed with bronchial asthma and allergic creativecommons.org/licenses/by/ rhinitis at the age of 3, hospitalized many times due to complaints of fatigue and persistent 4.0/). drug-resistant dyspnea—controlled therapy with inhaled corticosteroids was done with Children 2021, 8, 589. https://doi.org/10.3390/children8070589 https://www.mdpi.com/journal/children Children 2021, 8, x FOR PEER REVIEW 2 of 5 Children 2021, 8, 589 2 of 5 drug-resistant dyspnea—controlled therapy with inhaled corticosteroids was done with unsatisfactory results, the girl was under asthma therapy for 12 years, with no significant change in the presenting complaints. The patient has never been intubated, she had no unsatisfactory results, the girl was under asthma therapy for 12 years, with no significant other diseases in the past. change in the presenting complaints. The patient has never been intubated, she had no At the age of 15 the medical examination showed impaired general condition, mixed other diseases in the past. At the agedyspnea of 15 the and medical noisy examination and weakened showed breathing. impaired Physical general examination condition, mixed was dys-otherwise unim- pnea and noisypressive. and weakened breathing. Physical examination was otherwise unimpressive. The laboratoryThe tests laboratory showed peripheraltests showed hypereosinophilia, peripheral hypereosinophilia, elevated serum IgGelevated levels— serum IgG lev- 18.20 g/L (referenceels—18.20 range g/L 5.4–16.1 (reference g/L), range elevated 5.4–16.1 serum g/L), IgG4 elevated levels—8.25 serum IgG4 g/L (referencelevels—8.25 g/L (refer- range 0.23–1.11ence g/L), range IgG4/IgG 0.23–1.11 ratio—0.45.g/L), IgG4/IgG Spirometry ratio—0.45. showed: Spirometry total showed: lung capacity total lung capacity (TLC) 99.2%;(TLC) forced 99.2%; vital capacity forced vital (FVC) capacity 77.8%; (FVC) forced 77.8%; expiratory forced volume expiratory (FEV1) volume 83.8%; (FEV1) 83.8%; FEV1/FVC 88.71%;FEV1/FVC low positive88.71%; low bronchial positive dilation bronchial test—FEV1 dilation fifteentest—FEV1 minutes fifteen after minutes salbu- after salbut- tamol inhalation—93.9%.amol inhalation Chest—93.9%. computed Chest tomography computed tomography (CT) scan did (CT) not showscan did any not patho- show any patho- logical findingslogical in the findings lungs. in Fibrobronchoscopy the lungs. Fibrobronchoscopy was performed—fibrous was performed stenosis—fibrous was stenosis was found at the levelfound of at the the lower level edgeof the of lower the thyroid edge of cartilage the thyroid ring cartil withage an infiltrationring with an area infiltration area (Figure1a). No(Figure pathological 1a). No changes pathological were changes found in were the other found parts in the of other the tracheobronchial parts of the tracheobronchial tree. A fine-needletree. A aspirationfine-needle biopsy aspiration was biopsy taken. was Having taken. in Having mind systemic in mind connectivesystemic connective tis- tissue diseasesue at disease the beginning at the beginning (particularly (particularly Wegener’s Wegener disease),’s disease), specific antibodyspecific antibody tests tests were were performedperformed (anti-ds (anti DNA,-ds pANCA, DNA, pANCA, cANCA, cANCA, C3, C4)—all C3, C4) in the—all reference in the reference range. An range. An im- immunohistologicalmunohistological examination examination with CD 138 with was CD done—lymphoplasmic 138 was done—lymphoplasmic inflammatory inflammatory in- infiltration, squamousfiltration, metaplasiasquamous withmetaplasia dysplastic with changes dysplastic were changes found, were more found, than 40% more of than 40% of lymphoplasmocyteslymphoplasmocytes marked with CDmarked 138 express with CD IgG4 138 (Figure express2a,b). IgG4 Immunoproliferative (Figure 2a,b). Immunoprolifera- hyper-IgG4 diseasetive hyper was-IgG4 diagnosed disease based was diagnosed on elevated based serum on IgG4, elevated IgG4/IgG serum ratioIgG4, > IgG4 0.4 /IgG ratio > and histological0.4 findings.and histological findings. (a) (b) Figure 1. EndoscopicFigure 1. view.Endoscopic (a) Initial view. view (—a)a Initial fibrous view—a stenosis fibrous at the level stenosis of the at thelower level edge of of the the lower thyroid edge cartilage of ring with an infiltrationthe thyroid area; cartilage(b) a fibrous ring ring with after an infiltration dilation with area; rigid (b) tube a fibrous number ring 6.5, after followed dilation by with unaffected rigid tube trachea and annular cartilage.number 6.5, followed by unaffected trachea and annular cartilage. Corticosteroid treatment was initiated, at first systemic, after that orally, reaching a maintenance dose of 8 mg methylprednisolone daily. Later on, rigid bronchoscopy with partial resection of the stenotic fibers was performed, achieving considerable dyspnea reduc- tion. During maintenance therapy three more control bronchoscopies were performed—a reduction of the stenotic ring and improved patency of the trachea were reported. After two years of treatment (at the age of 17), the girl lacked subjective complaints— showed no shortness of breath or limitation of physical activity. The control spirometry was satisfactory: FVC—91%, FEV1—88%; normal blood acid-base state. The last fibro- bronchoscopy showed a fibrous ring, through which a 4.4 mm fibrobronchoscope passed, followed by an unaffected trachea and annular cartilage. Dilation of the fibrous area was performed with rigid tube number 6.5 (Figure1b). Children 2021, 8, x 589 FOR PEER REVIEW 3 of 5 (a) (b) Figure 2. HistopathologicHistopathologic findings findings in
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