Plasminogen Deficiency Research Articles

LİGNÖZ PERİODONTİTİS : OLGU SUNUMU

Aim: Ligneous periodontitis is an infectious disease characterized by the inflammation of the supporting periodontal tissues, resulting in attachment and alveolar bone loss with plasminogen deficiency. This case report shows the treatment and follow-up of patient who have ligneous periodontitis. Case Report: Fifteen years old boy who had ligneous periodontitis applied to our periodontology clinic. Clinical and radiographic evaluation revealed severe periodontal breakdown around all teeth. Intraoral soft tissues were characterized by gingival reddening and swelling and the patient complained of periodic discharge of pus from the periodontal pockets, mobility, and severe pain. The probing depths were more than 7 mm %58 percent probing areas and there were radiolucent lesions around the teeth in panoramic radiograph. Full-mouth subgingival scaling and root planing under local anesthesia was performed and the pocket wall is gently retracted with a periodontal curette in an attempt to initiate drainage through the pocket entrance. Systemic amoxicillin and clavulanic acid applied for antibiotic therapy. Result: The goal of treatment was to create a clinical condition that is conducive to retaining as many teeth as possible for as long as possible but alveolar bone loss around the teeth was too severe. Systemic disorders such as plasminogen deficiency increase a patient's susceptibility to destructive periodontitis and have impacts on periodontal disease progression and severity. Key Words: Ligneous periodontitis, plasminogen deficiency, systemic diseases, alveolar bone loss

Periodontitis associated with plasminogen deficiency: a case report

BackgroundPlasminogen deficiency is a rare autosomal recessive disease, which is associated with aggressive periodontitis and gingival enlargement. Previously described treatments of plasminogen deficiency associated periodontitis have shown limited success. This is the first case report indicating a successful therapy approach consisting of a non-surgical supra- and subgingival debridement in combination with an adjunctive systemic antibiotic therapy and a strict supportive periodontal regimen over an observation period of 4 years.Case presentationThe intraoral examination of a 17-year-old Turkish female with severe plasminogen deficiency revealed generalized increased pocket probing depths ranging from 6 to 9 mm, bleeding on probing over 30%, generalized tooth mobility, and gingival hyperplasia. Alveolar bone loss ranged from 30% to 50%. Clinical attachment loss corresponded to pocket probing depths. Aggregatibacter actinomycetemcomitans, Porphyromonas gingivalis, Treponema denticola, Prevotella intermedia, Prevotella nigrescens and Eikenella corrodens have been detected by realtime polymerase chain reaction. Periodontal treatment consisted of full mouth disinfection and adjunctive systemic administration of amoxicillin (500 mg tid) and metronidazole (400 mg tid). A strict supportive periodontal therapy regimen every three month in terms of supra- and subgingival debridement was rendered. The reported therapy has significantly improved periodontal health and arrested disease progression. Intraoral examination at the end of the observation period 3.5 years after non-surgical periodontal therapy showed generalized decreased pocket probing depths ranging from 1 to 6 mm, bleeding on probing lower 30%, and tooth mobility class I and II. Furthermore, microbiological analysis shows the absence of Porphyromonas gingivalis, Prevotella intermedia and Treponema denticola after therapy.ConclusionAdjunctive antibiotic treatment may alter the oral microbiome and thus, the inflammatory response of periodontal disease associated to plasminogen deficiency and diminishes the risk of pseudomembrane formation and progressive attachment loss.This case report indicates that patients with plasminogen deficiency may benefit from non-surgical periodontal treatment in combination with an adjunctive antibiotic therapy and a strict supportive periodontal therapy regimen.

The Tissue Fibrinolytic System Contributes to the Induction of Macrophage Function and CCL3 during Bone Repair in Mice

Macrophages play crucial roles in repair process of various tissues. However, the details in the role of macrophages during bone repair still remains unknown. Herein, we examined the contribution of the tissue fibrinolytic system to the macrophage functions in bone repair after femoral bone defect by using male mice deficient in plasminogen (Plg –/–), urokinase-type plasminogen activator (uPA –/–) or tissue-type plasminogen activator (tPA –/–) genes and their wild-type littermates. Bone repair of the femur was delayed in uPA –/– mice until day 6, compared with wild-type (uPA +/+) mice. Number of Osterix-positive cells and vessel formation were decreased in uPA –/– mice at the bone injury site on day 4, compared with those in uPA +/+ mice. Number of macrophages and their phagocytosis at the bone injury site were reduced in uPA –/– and Plg –/–, but not in tPA –/– mice on day 4. Although uPA or plasminogen deficiency did not affect the levels of cytokines, including TNF-α, IL-1β, IL-6, IL-4 and IFN-γ mRNA in the damaged femur, the elevation in CCL3 mRNA levels was suppressed in uPA –/– and Plg –/–, but not in tPA –/– mice. Neutralization of CCL3 antagonized macrophage recruitment to the site of bone injury and delayed bone repair in uPA +/+, but not in uPA –/– mice. Our results provide novel evidence that the tissue fibrinolytic system contributes to the induction of macrophage recruitment and CCL3 at the bone injury site, thereby, leading to the enhancement of the repair process.

Response of Plasminogen Deficiency Associated Ligneous Conjunctivitis to Topical Fresh frozen Plasma with Heparin

Introduction: Ligneous conjunctivitis is rare form of conjunctivitis characterized by woody fibrinous membranes. Plasminogen deficiency has been introduced as the most common cause and formation of membrane is the result of impaired wound healing process. Case Report: We present a 4-month-old male infant with chronic membranous conjunctivitis. Multiple scrapping of membranes were performed with no improvement. Fibrinous membranes were also seen on nasal cavity and upper gingiva. After his blood revealed plasminogen blood levels of 11.9% (normal: 75-135%), diagnosis of ligneous conjunctivitis due to plasminogen deficiency was obtained. Treatment was then changed to topical fresh frozen plasma and heparin. Dramatic resolution of membranes was observed after 15 months therapy combined with membranes scraping. Conclusion: Ligneous conjunctivitis is rare manifestation of plasminogen deficiency that occurs sporadically where local infection and multiple surgeries might act as trigger to induce fibrinous membranes. Topical heparin and FFP could treat the disease successfully.

Ligneous conjunctivitis: A rare case report

Ligneous conjunctivitis is a rare condition characterized by the formation of a pseudomembrane on tarsal conjunctiva and at some other sites including mucous membranes in mouth, nasopharynx, vagina etc., Ligneous conjunctivitis may be inherited in an autosomal recessive pattern due to mutations in the plasminogen gene and a variety of other genes, associated with type I plasminogen deficiency (hypoplasminogenemia), leading to impaired wound healing. We are presenting a case of 35 years old female suffering from ligneous conjunctivitis which was removed surgically.

Root dentin anomaly and a PLG mutation

We report a Thai girl affected with plasminogen deficiency, Type I. Ligneous conjunctivitis was first observed when she was one-month-old. The newly recognized findings include tapered incisor roots as a result of thin root dentin, generalized short tooth roots, and mandibular prognathism. Mutation analysis of PLG demonstrated homozygous c.1193G>A missense mutation. The parents were heterozygous for c.1193G>A mutation. The c.1193G>A mutation is novel and predicted to cause amino acid substitution p.Cys398Tyr. Thin root dentin in the patient who was affected with PLG mutation and immunolocalization of Plg during early root development in mice imply the role of plasminogen in root dentin formation.

Plasminogen Is a Joint‐Specific Positive or Negative Determinant of Arthritis Pathogenesis in Mice

A fundamental metric in the diagnosis of arthropathies is the pattern of joint involvement, including differences in proximal versus distal joints and patterns of symmetric or asymmetric disease. The basis for joint selectivity among arthritides and/or within a defined disease such as rheumatoid arthritis remains enigmatic. Coagulation and fibrinolytic activity are observed in both experimental animals with inflammatory joint disease and patients with inflammatory arthritis. However, the contribution of specific hemostatic factors to joint disease is not fully defined. We sought to determine the contribution of the fibrinolytic protease, plasminogen, to tumor necrosis factor α (TNFα)-driven arthritis in distinct joints in mice. The impact of plasminogen and/or fibrinogen genetic deficiencies on arthritis progression was evaluated in Tg197 mice genetically predisposed to spontaneous, nonabating, and erosive polyarthritis due to exuberant human TNFα expression. Elimination of plasminogen in Tg197 mice significantly exacerbated the incidence and severity of arthritis within the paw joints, but simultaneously and dramatically diminished the entire spectrum of pathologies within the knee joints of the same animals. These opposing outcomes were both mechanistically linked to fibrin(ogen), in that superimposing fibrinogen deficiency reversed both the proarthritic phenotype in the paws and arthritis resistance in the knees of plasminogen-deficient mice. Intriguingly, the change in disease severity in the knees, but not the paws, was associated with a plasminogen-dependent reduction in matrix metalloproteinase 9 activity. Plasminogen is a key molecular determinant of inflammatory joint disease capable of simultaneously driving or ameliorating arthritis pathogenesis in distinct anatomic locations in the same subject.

Plasminogen deficiency causes reduced corticospinal axonal plasticity and functional recovery after stroke in mice.

Tissue plasminogen activator (tPA) has been implicated in neurite outgrowth and neurological recovery post stroke. tPA converts the zymogen plasminogen (Plg) into plasmin. In this study, using plasminogen knockout (Plg-/-) mice and their Plg-native littermates (Plg+/+), we investigated the role of Plg in axonal remodeling and neurological recovery after stroke. Plg+/+ and Plg-/- mice (n = 10/group) were subjected to permanent intraluminal monofilament middle cerebral artery occlusion (MCAo). A foot-fault test and a single pellet reaching test were performed prior to and on day 3 after stroke, and weekly thereafter to monitor functional deficit and recovery. Biotinylated dextran amine (BDA) was injected into the left motor cortex to anterogradely label the corticospinal tract (CST). Animals were euthanized 4 weeks after stroke. Neurite outgrowth was also measured in primary cultured cortical neurons harvested from Plg+/+ and Plg-/- embryos. In Plg+/+ mice, the motor functional deficiency after stroke progressively recovered with time. In contrast, recovery in Plg-/- mice was significantly impaired compared to Plg+/+ mice (p<0.01). BDA-positive axonal density of the CST originating from the contralesional cortex in the denervated side of the cervical gray matter was significantly reduced in Plg-/- mice compared with Plg+/+ mice (p<0.05). The behavioral outcome was highly correlated with the midline-crossing CST axonal density (R2>0.82, p<0.01). Plg-/- neurons exhibited significantly reduced neurite outgrowth. Our data suggest that plasminogen-dependent proteolysis has a beneficial effect during neurological recovery after stroke, at least in part, by promoting axonal remodeling in the denervated spinal cord.

Lp(a)/apo(a) Modulate MMP-9 Activation and Neutrophil Cytokines in Vivo in Inflammation to Regulate Leukocyte Recruitment

Lipoprotein(a) [Lp(a)] is an independent risk factor for cardiovascular diseases, but the mechanism is unclear. The pathogenic risk of Lp(a) is associated with elevated plasma concentration, small isoforms of apolipoprotein [apo(a)], the unique apolipoprotein of Lp(a), and a mimic of plasminogen. Inflammation is associated with both the initiation and recovery of cardiovascular diseases, and plasminogen plays an important role in leukocyte recruitment. Because Lp(a)/apo(a) is expressed only in primates, transgenic mice were generated, apo(a)tg and Lp(a)tg mice, to determine whether Lp(a)/apo(a) modifies plasminogen-dependent leukocyte recruitment or whether apo(a) has an independent role in vivo. Plasminogen activation was markedly reduced in apo(a)tg and Lp(a)tg mice in both peritonitis and vascular injury inflammatory models, and was sufficient to reduce matrix metalloproteinase-9 activation and macrophage recruitment. Furthermore, neutrophil recruitment and the neutrophil cytokines, CXCL1/CXCL2, were suppressed in apo(a)tg mice in the abdominal aortic aneurysm model. Reconstitution of CXCL1 or CXCL2 restored neutrophil recruitment in apo(a)tg mice. Apo(a) in the plasminogen-deficient background and Lp(a)tg mice were resistant to inhibition of macrophage recruitment that was associated with an increased accumulation of apo(a) in the intimal layer of the vessel wall. These data indicate that, in inflammation, Lp(a)/apo(a) suppresses neutrophil recruitment by plasminogen-independent cytokine inhibition, and Lp(a)/apo(a) inhibits plasminogen activation and regulates matrix metalloproteinase-9 activation and macrophage recruitment.

Comprehensive Genetic Analysis of Complement and Coagulation Genes in Atypical Hemolytic Uremic Syndrome

非典型溶血性尿毒症症候群（aHUS）は，細胞表面における補体第二経路の制御不能な活性化等による血栓性微小血管障害に起因し，微小血管性溶血性貧血・血小板減少・急性腎不全を引き起こす。患者の約半数で，補体制御・活性化因子の遺伝子異常が見出され，その病態における補体系の関与が示唆されている。一方本症の原因としては，補体系以外の要因の存在も疑われている。著者らは，散発性aHUS 患者36 人を対象に，ターゲットエンリッチメント・次世代シーケンスにより，補体系に加え凝固系経路の遺伝子異常に関する包括的スクリーニングを行った。その結果，計84 の非同義的バリアントが見出されたが，うち22 については既報告のものであった。コンピュータ解析により，残る62 のうち20 のバリアントが有害性と推測された。また，42 のうち約半数の19（45％）のバリアントは，本症の病因との関連がこれまでに示された遺伝子に存在した。また新たな知見として，凝固系のいくつかの遺伝子が，病因として重要なことが判明した。特に，PLG 遺伝子の異常が，aHUS の病態に関連深いことが示された。

Comprehensive Genetic Analysis of Complement and Coagulation Genes in Atypical Hemolytic Uremic Syndrome

Atypical hemolytic uremic syndrome (aHUS) is a thrombotic microangiopathy caused by uncontrolled activation of the alternative pathway of complement at the cell surface level that leads to microangiopathic hemolytic anemia, thrombocytopenia, and acute kidney failure. In approximately one half of affected patients, pathogenic loss-of-function variants in regulators of complement or gain-of-function variants in effectors of complement are identified, clearly implicating complement in aHUS. However, there are strong lines of evidence supporting the presence of additional genetic contributions to this disease. To identify novel aHUS-associated genes, we completed a comprehensive screen of the complement and coagulation pathways in 36 patients with sporadic aHUS using targeted genomic enrichment and massively parallel sequencing. After variant calling, quality control, and hard filtering, we identified 84 reported or novel nonsynonymous variants, 22 of which have been previously associated with disease. Using computational prediction methods, 20 of the remaining 62 variants were predicted to be deleterious. Consistent with published data, nearly one half of these 42 variants (19; 45%) were found in genes implicated in the pathogenesis of aHUS. Several genes in the coagulation pathway were also identified as important in the pathogenesis of aHUS. PLG, in particular, carried more pathogenic variants than any other coagulation gene, including three known plasminogen deficiency mutations and a predicted pathogenic variant. These data suggest that mutation screening in patients with aHUS should be broadened to include genes in the coagulation pathway.

Factors associated with the development of superficial vein thrombosis in patients with varicose veins

IntroductionSuperficial vein thrombosis (SVT) is a common and controversial clinical entity. Recent studies have demonstrated that SVT should be seen as a venous thromboembolism (VTE). The objective of this study was to investigate the prevalence of thrombophilia defects and to estimate the role of age, sex and body mass index (BMI) in patients with varicose veins (VVs) and SVT. Materials and MethodsA total of 230 patients with VVs, 128 with, and 102 without SVT underwent thrombophilia testing included factor V Leiden, prothrombin G20210A, methylenetetrahydrofolate reductase and plasminogen activator inhibitor- 1 mutations, protein C, protein S (PS), anti-thrombin III and plasminogen deficiencies and levels of A2 antiplasmin, activate protein C resistance and lupus anticoagulant. According to Clinical- Etiology- Anatomy- Pathophysiology (CEAP) classification patients were categorized in two subgroups: moderate disease (C2,3) and severe disease (C4,5,6). Age and body mass index were also assessed. ResultsThe prevalence of thrombophilia defects was significantly higher in patients with moderate disease and SVT (p=0.002). In the C2,3 group, SVT was associated with PS deficiency (p=0.018), obesity (p<0.001), male gender (p=0.047) and age (p<0.001). There were no significant differences in patients with severe disease. ConclusionsAge, male sex, obesity and PS deficiency are factors associated with SVT development among patients with VVs having moderate disease (C2,3).

Conjunctival instillation of plasminogen eliminates ocular lesion in B6.129P2-Plgtm1Jld transgenic mice, a model of ligneous conjunctivitis

Ligneous conjunctivitis is a severe and rare chronic “idiopathic membraneous” conjunctivitis, characterized by the formation of pseudomembranes mostly on the palpebral surfaces that progressively replace the normal mucosa. Evidence has been provided that ligneous conjunctivitis is caused by a severe systemic plasminogen deficiency with decreased plasminogen antigen and decreased plasminogen functional activities. Objective of the present study is to verify the hypothesis that a topical eye application of plasminogen is able to ameliorate the consequences of this disease. Here we report the results of pre-clinical studies performed to investigate the therapeutic effectiveness of an eye-drop plasminogen preparation in B6.129P2-Plgtm1Jld transgenic mice, a model of ligneous conjunctivitis.The entity of protection mediated by plasminogen was evaluated by measuring the extent of the eye lesion by means of a computerized system and dedicated software.The results of the present study clearly showed that the administration for six times a day of plasminogen eye-drop solution in the lesioned eye of animals knock-out for plasminogen gene and developing ligneous conjunctivitis caused a dose and time related reduction of the extent of the ocular lesion. These findings may pave the road for the pharmacological treatment of the ocular lesion associated to the ligneous conjunctivitis that at the present is surgically treated by removing the pseudomembranes generated on the eye.

重度プラスミノゲン欠損症における凝固線溶反応の変容; 血栓溶解における代替的線溶経路の意義(仮説)

重度プラスミノゲン欠損症における凝固線溶反応の変容; 血栓溶解における代替的線溶経路の意義(仮説)

Ligneous Cervicitis in a Woman With Plasminogen Deficiency Associated With an Atypical Form of Microglandular Hyperplasia

A 19-yr-old woman with previously diagnosed clear cell adenocarcinoma was referred to the Charité for further treatment. Biopsies were taken from the cervix, the endometrium, pseudomembranes in the peritoneum, and sentinel lymph nodes. The morphologic picture of pseudomembranes and inflammation together with the provided information about plasminogen deficiency of the patients led to the hypothesis of ligneous cervicitis. The previously taken biopsies of the adenocarcinoma were reevaluated and showed a clear cell lesion. Further immunohistochemical examination with antibodies against p16, Ki67, CEA, and p53 could not prove its malignant character. As a result we diagnosed an atypical form of microglandular hyperplasia in a patient with ligneous cervicitis. Ligneous cervicitis is a rare inflammatory condition that might affect all mucus membranes in patients with plasminogen deficiency. This case shows the importance of correlating pathologic and clinical findings in the diagnosis of ligneous cervicitis because of the rarity of the disease and the heterogeneity at presentation.

The ventriculocholecystic shunt: two case reports and a review of the literature

We describe the re-siting of ventriculoperitoneal shunts to the gallbladder in two children. The first child had a rare case of hydrocephalus associated with plasminogen deficiency. She had had multiple VP shunt revisions due to non-absorption of CSF from the peritoneum. The second had craniopharyngioma-related hydrocephalus with once again a non-absorbing peritoneum. We report no surgical complications in the revisions for both the cases, and there has been a subsequent follow-up of 46 and 28 months, respectively, without incident. A review of the relevant literature describing the use and the performance of ventriculocholecystic shunts in comparison with other ventricular shunts is considered.

Abstract WMP67: Plasminogen Deficiency And Gender Dimorphism After Thromboembolic Stroke

Background: Stroke is a sex dimorphic disease with males and post-menopausal females having worse outcomes. Plasminogen (Pg) system plays a major role in post-stroke outcomes, affecting clot lysis and MMP-9 regulation. Activation of MMP-9 occurs by both Pg-dependent and Pg-independent mechanisms. However, the role of Pg-dependent molecular pathways in stroke remains unclear. The aim of this work was to determine if there is a sex-specific change in post-stroke neurologic outcomes in mice with Pg deficiency. Methods: Pg knockout (Pg -/- ) and wild type (Pg +/+ ) mice of both sexes (n=8-14 animals/group; 26±4 weeks old) were subjected to embolic stroke. At 24 hours, behavioral evaluation, infarct area and edema volume were assessed. Results: Plasminogen deficiency caused pathological fibrin deposition in the microcirculation, inducing a similar systemic inflammatory response in males and females with significantly increased levels of monocytes (1.7-fold, p&lt;0.01) and granulocytes (3.5-fold, p&lt;0.001) in the circulation compared with Pg +/+ mice. Moreover, all pro-thrombotic Pg -/- mice had microcytic anemia due to low red blood cell volume (36.7 fl vs. 41.6 fl, p&lt;0.001). Pg -/- females had lower estrogen levels compared with Pg +/+ females (10.2±12.6 pg/ml vs. 31.5±51 pg/ml; p=0.23). Pg deficiency did not affect the level of MMP-9 in the brain nor in the circulation. Despite the similar levels of systemic pro-thrombotic inflammation in Pg -/- mice of both sexes, we observed a significant gender disparity in stroke outcomes. Pg deficiency in males improved behavioral outcomes (1.6 vs. 3.2; p&lt;0.001), reduced infarct size (32.8% vs. 43.1%; p&lt;0.05) and edema volume (5.7% vs. 15.4%; p&lt;0.001) compared with Pg -/- mice. On the other hand, Pg -/- females had a higher mortality (60% vs. 18%) and a poorer neurologic score (4.6 versus 2.3; p&lt;0.001) due to severe brain injury with an increased volume of infarct (64.8% vs. 19.9%; p&lt;0.001) and edema (13.3% vs. 5.6%; p&lt;0.05) compared with Pg +/+ females. Conclusion: Unlike males, Pg -/- females had disproportionately poor outcomes compared with Pg +/+ females. Our data demonstrates that Pg -/- female mice may represent an experimental model that mimics females in an early menopausal stage with chronic pro-thrombotic disease.

Plasmin‐driven fibrinolysis facilitates skin tumor growth in a gender‐dependent manner

Rearrangement of the skin during wound healing depends on plasmin and plasminogen, which serve to degrade fibrin depositions in the provisional matrix and thereby facilitate keratinocyte migration. In the current study, we investigated whether plasmin and plasminogen likewise played a role during the development of skin cancer. To test this, we set up a chemically induced skin tumor model in a cohort of mice and found that skin tumor growth in Plg(-/-) male mice was reduced by 52% compared with wild-type controls. Histological analyses suggested that the growth-restricting effect of plasminogen deficiency was due to thrombosis and lost patency of the tumor vasculature, resulting in tumor necrosis. The connection between plasmin-dependent fibrinolysis, vascular patency, and tumor growth was further substantiated as the effect of plasminogen deficiency on tumor growth could be reverted by superimposing heterozygous fibrinogen deficiency on Plg(-/-) mice. Tumors derived from these Fib(-/+);Plg(-/-) mice displayed a significantly decreased level of tumor thrombosis compared with Plg(-/-) mice. In summary, these data indicate that plasmin-driven fibrinolysis facilitates tumor growth by maintaining patency of the tumor vasculature.

Destructive Membranous Periodontal Disease (Ligneous Gingivitis): A Literature Review

Destructive membranous periodontal disease, or ligneous gingivitis, is a rare condition involving nodular gingival enlargement with ulceration and periodontal tissue destruction. This review gives a brief account of the cases reported in the literature. An effort is also made to define the periodontal disease caused by plasminogen deficiency with a view to its potential inclusion in the classification of periodontal diseases. A MEDLINE/PubMed and manual search was conducted to find papers describing ≥1 case of ligneous disease involving the oral mucosa. We identified 23 articles reporting 35 cases. For each patient, we analyzed various characteristics, including age, sex, age of onset, oral symptoms, histologic features, plasminogen levels (functional activity, plasma antigen), genetic features, treatment, and results of treatment during the reported follow-up. Ligneous gingivitis is a rare periodontal disorder closely associated with ligneous conjunctivitis and plasminogen deficiency. Its diagnosis may be supported by the finding of genetic mutations responsible for the condition. Research is focusing on the future development of an effective therapy capable of arresting the destructive evolution of the disease. Additional studies, investigating features such as probing depth and attachment loss, are needed for the appropriate classification of this periodontal disease.

Laryngeal obstruction in congenital plasminogen deficiency

Type 1 congenital plasminogen deficiency (CPD) is a rare autosomal recessive disease which causes formation of fibrin pseudomembranes that affect multiple systems/organs: the eyes, respiratory system, urinary and genital systems, gastrointestinal system, and the central nervous system. We present a rare manifestation of the disease-severe upper airway obstruction due to a rapidly growing mass in the supraglottic region-6 months after dental treatment under general anesthesia. The management of such a manifestation has not been discussed in the current literature. Due to deterioration in his clinical status, the patient eventually underwent both a tracheotomy in order to bypass the obstruction, and excision of the supraglottic mass. Within a few days the mass recurred with complete obstruction of the upper airway.