Rare Late Complication Research Articles

Metastatic renal cell carcinoma presenting as gastrointestinal bleeding.

A 73-year-old female with metastatic renal cell carcinoma (RCC) presented with melena and lethargy. She was found to be iron deficient with a hemoglobin of 101 g/L. Her gastroscopy found six irregular pedunculated non-bleeding polyps, 10-20 mm in diameter. Histopathology of the resected polyp returned as metastatic clear cell renal cell carcinoma. Gastric metastases from any primary malignancy are rare and metastatic RCC accounts for only 7% of these tumors. Furthermore, while RCC commonly metastasises to the lung, bone and lymph nodes, metastasis to the gastrointestinal tract is extremely rare, occurring in <1% of patients. Presentation of RCC as a gastric polyp is usually a late event, and on average occurs 6.7 years after initial diagnosis of RCC. Therefore, this case highlights a rare but important late complication of RCC, presenting as gastrointestinal bleeding secondary to gastric metastases.

Bilateral dead bag syndrome

Introduction: Posterior capsular rupture (PCR) is defined as breach in the continuity of the posterior part of the capsular bag in the human crystalline lens. Dead bag syndrome (DBS), a term coined by Samuel Masket, is a rare late complication of cataract surgery, which may present with spontaneous posterior capsular tear, causing intraocular lens (IOL) decentration or dislocation. It is usually seen in patients who have undergone an uneventful phacoemulsification with in-the-bag posterior chamber IOL implantation many years ago. Patient and Clinical Findings: A 73-year-old man presented with reduced vision in the right eye 17 years after an uneventful phacoemulsification and in-the-bag posterior chamber IOL implantation. There was no history of trauma to the eye. On examination, a decentered IOL with an inferior PCR was noted. Diagnosis, Intervention, and Outcomes: Decentered IOL was explanted, and a retro-iris–fixated IOL was implanted. Similar findings were noted in the left eye 4 years after the involvement of the right eye. There was no IOL dislocation or decentration. A diagnosis of DBS as the cause of PCR was made. The left eye was managed conservatively, and the patient remains under observation. Conclusions: The case described here seems to correspond to bilateral DBS. Physicians should be alert to look for the possibility of development of a similar complication in the other eye.

Systematic application of SICA-PED protocol for central venous catheterization in neonates: A prospective clinical study on 104 cases.

Catheterization of central vessels can be associated with early and late, potentially fatal complications. A proactive approach is imperative to reduce the frequency and magnitude of adverse events. Recently, the GAVeCeLT has proposed a protocol called SICA-PED (i.e. Safe Insertion of Central Access in Pediatric patients) and includes seven evidence-based strategies. Through a single-center prospective observational study, the authors wanted to consolidate the efficacy and safety of these protocol in newborns. In a series of 104 newborns, the seven steps of the protocol were applied (1) pre-procedural ultrasound study of the RaCeVA veins, (2) correct aseptic technique, (3) ultrasound-guided venipuncture, (4) intraprocedural localization of the tip of the catheter with TTE (ECHO TIP) and (iECG) intracavitary electrocardiogram, (5) reasoned choice of the implant exit site with the RAVESTO Tunneling technique, (6) anchoring without stitches, and (7) exit point protection with the use of glue and transparent semipermeable membrane. The authors have included a further precaution in point (6) the subcutaneous anchoring system has added the counter-fixation of the catheter wings that we will call 6Plus Point. All infants requiring implantation of elective us-guided central venous access were enrolled in the study. None of the 104 implanted central venous catheters experienced early complications (accidental arterial puncture, PNX, primary malposition); rare late complications such as ecchymosis, CRBSI, exit site infection or dislodgement were observed, No catheter-related thrombotic phenomena were observed. The CRBSI catheter-related infection rate was 2.47 × 1000 days catheter cases. The results of this prospective study strengthen the feasibility and efficacy of the SICA-Ped Protocol. Demonstrating that the systematic application of the evidence-based seven-step implantation strategy increases the success rate, minimizes early and late complications, which result in increased patient safety.

Endotension Following Endovascular Aneurysm Repair: Retrospective Review of Treatment and Clinical Outcome.

: Endotension is a rare late complication characterized by an increase in sac size without any type of endoleak following endovascular aortic aneurysm repair (EVAR). Due to its rarity, few studies have demonstrated the mechanism behind and the management of endotension. In this study, we aimed to better understand the treatment and the long-term outcome of endotension in a single-center cohort. : This study was designed for a retrospective review of the patients diagnosed with endotension between January 2006 and December 2017. The study patients were categorized into two groups (primary versus secondary) based on the presence of any type of endoleak before the diagnosis of endotension. We collected data related to endotension treatment, intraoperative findings, and long-term outcomes. : In a cohort of 15 patients diagnosed with endotension following EVAR, eight were classified into the primary endotension (PE) group without prior endoleak, and seven exhibited secondary endotension (SE). Among the eight PE patients, endovascular intervention for a preemptive purpose was conducted in six patients; however, three (50%) showed continuous sac expansion and finally received open conversion. Overall, eight patients (five in PE and three in SE) underwent open conversion, and one (12.5%) presented with an undetected endoleak during the operative findings. Postoperative morbidity was observed in three patients with no operative mortality. : Endotension can be managed initially through simple observation for changes on serial images, along with preemptive endovascular intervention. However, surgical intervention should be considered for patients with specific indications including continuous aneurysm sac enlargement, presence of symptoms, suspicions of migration of stent-graft with endoleak, and infection.

You’re a pain in my side! Abscess and microperforation as a complication of therapy from early-stage endometrial cancer: A case report

This paper reports on a 55-year-old woman presenting with left lower quadrant and groin pain that posed a significant diagnostic challenge. She had a history of obesity and stage 1B endometrial carcinoma treated with surgery and radiation 1-year prior. Despite several unsuccessful biopsy attempts and unclear imaging findings, she was ultimately diagnosed with a pelvic sidewall abscess secondary to a bowel microperforation, a rare late complication of radiation related to adhesions, weakened bowel, and peristalsis. Her condition was successfully treated with drainage and antibiotics. It is widely known that patients with endometrial cancer and comorbid obesity often experience diagnostic delay, weight stigma, and other barriers and thus deserve careful attention and continued advocacy.

Very late amplatzer device thrombosis and stroke

In many large center randomized controlled studies, it has been shown that percutaneous closure is more effective in patients with Atrial Septal Defect (ASD) who have cryptogenic stroke, when percutaneous closure is compared with medical treatment [1-6]. However, thrombus formation in percutaneous closure devices is one of the known and rare late complications [7]. In this context, we present a case of recurrent stroke attack after late thrombus development in the ASD closure device in a patient receiving dual antithrombotic therapy in our clinic

Therapy-Related Acute Myeloid Leukemia After Chemotherapy For Osteosarcoma: Report of 2 Cases and Review of The Literature

Abstract Introduction/Objective Therapy-related acute myeloid leukemia (t-AML) and myelodysplastic syndrome (t-MDS) are rare but well-established late complications of chemotherapeutic agents or radiation therapy used to treat various malignancies. Many cases of t-AML/t-MDS have been described secondary to Hodgkin, non-Hodgkin lymphoma, and other solid tumors. However, only a few cases of t-AML and t-MDS have been reported following osteosarcoma treatment. We describe two patients diagnosed with osteosarcoma who developed t-AML after receiving multiple chemotherapeutic agents. Methods/Case Report A 12-year-old male with Li-Fraumeni syndrome was diagnosed with osteosarcoma of the left ilium and sacrum with pulmonary metastases. Despite multiple lines of chemoradiation therapy including doxorubicin and ifosfamide, tumor progressed at the primary tumor site. The patient subsequently presented with altered mental status and pancytopenia. Bone marrow biopsy demonstrated AML. He died despite supportive care. A 14-year-old female was diagnosed with left tibial osteosarcoma with bony and pulmonary metastases. She was treated on standard osteosarcoma chemotherapy including doxorubicin, cisplatin, methotrexate, ifosfamide and etoposide, surgery, and radiation. 1 year following therapy, she had local recurrence of osteosarcoma without progression despite not starting chemotherapy. However, 3 years later, she developed pancytopenia, and immature white blood cells were identified on peripheral smear. Bone marrow biopsy revealed AML-M7. Despite supportive care and zoledronic acid, she ultimately died from sepsis and disseminated intravascular coagulation. Results (if a Case Study enter NA) NA Conclusion With improved therapy and survival for osteosarcoma patients, these patients should be monitored for secondary malignancies. T-AML is rare following osteosarcoma treatment, but outcome remains poor, despite treatment. Underlying Li-Fraumeni syndrome may influence the risk of development of secondary malignant neoplasms. Literature review of types and prognosis of t-AML following osteosarcoma will be reviewed and compared to our cases, including related genetic predisposing factors and chemotherapeutic agents. Our second patient is the first reported case of AML-M7 secondary to osteosarcoma treatment.

Stump Appendicitis: A Rare Late Complication of Appendectomy, a Retrospective Analysis of 9082 Appendectomy Cases

Objective: Appendectomy for appendicitis is one of the most common surgical procedures performed worldwide. The remnant of the appendix stump after the first appendectomy carries the risk of developing stump appendicitis. Stump appendicitis is a rare late complication of appendectomy; inflammation occurs in the remaining appendicular stump. Delayed diagnosis of this condition can cause serious complications. Stump appendicitis is indeed a recognized clinical picture, but is often overlooked when evaluating patients with right lower quadrant abdominal pain, especially those with a history of appendectomy. It remains a clinical challenge because of its often delayed diagnosis and effective treatment, and possible accompanying morbidity or mortality. Material and Methods: Materials and Methods: We retrospectively screened the patients who were hospitalized in our general surgery clinic and diagnosed with stump appendicitis in the hospital for 12 years. There were 11 patients between January 2011 and 2023 Results: The mean age of all 11 patients described was 55 years (range: 20 to 66). 72% of the patients were male (8/11 males and 3/11 females). 63% of the patients had their first operations open, and 36% of them were laparoscopic. The mean white blood cell count on presentation of all reported 11 cases was 11,996 cells/mm3 (range: 5930 to 18,740), the mean fever was 37.82°C (range: 36.8 to 38.6), and the mean CRP count on presentation of all cases was 36.7 (range: 0.4 to 142.91). The most commonly performed radiographic examination used to diagnose stump appendicitis is the abdominal computerized tomography (CT) scan. It was used in 100% (11 cases). Ultrasound was also used in 100% (11 cases). Conclusion: The appendicular base must be accessed prior to undertaking an appendectomy, irrespective of the chosen strategy or technique. It is noteworthy that, apart from open or laparoscopic appendectomy, antibiotic therapy should also be considered as part of the treatment regimen, as documented in the literature series.

Postrhinoplasty Nasal Radix Cyst Treated with Endoscopic Marsupialization.

Postrhinoplasty nasal radix cyst is a rare late complication, with few cases being reported in the literature. The appropriate treatment for this cyst is surgical removal. To date, an open approach or intracartilagenous incision has been performed in most cases. Herein, we present a case of a 68-year-old man with a bulging mass of radix who visited our clinic. The patient underwent open surgery for the mass, but the mass recurred 2 years after surgery. Although removal using the open approach was planned as in the previous surgery, marsupialization was performed because a connection with the frontal recess was seen on a computed tomography scan. We report a rare case of nasal radix cyst successfully treated with marsupialization without an external scar.

Retiform endothelial hyperplasia mimicking cavernous malformation as a late complication of Gamma Knife radiosurgery.

Gamma Knife radiosurgery (GKRS) is a powerful tool for the management of arteriovenous malformations; however, newly formed mass lesions resembling cavernous malformations are a rare late complication of GKRS. In this retrospective study, the authors tried to clarify the unique histological features of these mass lesions. The authors retrospectively reviewed the clinical course of 889 patients who had undergone GKRS for arteriovenous malformations at their institute from 1991 to 2021. Among the 848 patients who had been followed up periodically with neuroradiological imaging, 37 developed a mass lesion mimicking a cavernous malformation and underwent surgical removal of the lesion. The median volume of the original nidus was 3.7 cm3 (range 0.07-30.5 cm3), and the median prescription dose was 21 Gy (range 12-25 Gy). The histological characteristics and radiological and clinical features of the 37 patients were investigated. Histological examination showed an organized hematoma and a structure termed "retiform endothelial hyperplasia" (RFEH) consisting of endothelium forming multiple lumen-like vascular channels mimicking cavernous malformations but lacking the subendothelial connective tissue that forms the typical vascular wall structure found in cavernous angioma and capillary telangiectasia. RFEH was detected a median of 10.8 years (range 3.2-27.4 years) after GKRS. Neuroimaging showed hematoma surrounded by massive brain edema in all 37 patients. Symptoms caused by mass effect of the lesion and perifocal edema worsened relatively rapidly but completely disappeared after surgery. No recurrence or morbidity occurred after the surgery. The delayed formation of RFEH that is mimicking a cavernous malformation neuroradiologically but is histologically distinct from a vascular malformation is a potential complication of GKRS. Its progressive clinical course suggests that surgical removal should be considered for symptomatic patients and/or patients with an apparent radiological mass sign.

Ureteroureteral Fistula as a Rare Complication of Ileal Conduit Urinary Diversion: a Case Report

Long-term studies on ileal conduit have shown a complication rate of up to 66%. Fistulas are a rare late complication of ileal conduit. To our knowledge, no data about ureteroureteral fistulas has been published yet. We present the case of a 73-year-old man with non-muscle invasive high-grade urothelial carcinoma of the bladder who failed to BCG due to intolerance and developed a cripple bladder. He underwent a cystoprostatectomy, pelvic lymphadenectomy, and an ileal conduit urinary diversion. Four months after the surgery, a raised creatinine level was discovered. Sonography revealed bilateral hydro-ureteronephrosis. Shortly after, the stomal urinary production became minimal, and a nephrostomy was introduced to the right kidney. As a result, the right- as well as the left-sided hydronephrosis diminished, as assessed by ultrasound. Through a loopogram and anterograde pyelography, a stenosis at the ureteroileal anastomosis as well as a connection between the two ureters was proven. As an endoscopic intervention to resolve the obstruction failed, a re-laparotomy to make a neo-anastomosis from the ureters to the ileal conduit was performed. We suspect that total obliteration of the oral end of the ileal conduit caused fistula formation of the ureters. We would like to address this rare phenomenon as a complication of ileal conduit to be mentioned in the differential diagnosis if the outcome of urine is in question. It is not a simple stenosis of the distal portion of the ureters and demands an adapted surgical reconstructing technique as well.

P113 TRANSCATHETER CORRECTION OF RESIDUAL INFERIOR SINUS VENOSUS DEFECT AFTER SURGICAL CLOSURE

Abstract A 21–year old male who underwent surgical closure of an isolated inferior type sinus venosus atrial septal defect (SV–ASD) at 6 years of age was admitted to our institute for acute superior limb hypotonia and dysarthria, with brain computed tomography scan positive for an ischaemic event. The contrast transcranial Doppler (CTD) was positive for right–to–left shunt (RLS). The transesophageal echocardiogram (TOE) revealed a small ASD (3.8 mm) near the inferior vena cava (IVC) with a small baseline left–to–right shunt and severe RLS after Valsalva’s Manovre (&amp;gt;20 bubbles) (fig.A–B). No clear intra–cardiac source of embolization was detected on workup. After a multidisciplinary discussion, percutaneous closure of residual ASD was planned. The main challenge was represented by inadequate inferior–posterior rim towards the IVC (&amp;lt;5 mm). A Figulla Flex II ASD Occluder with the smallest waist of 4 mm was implanted with no evidence of residual intraprocedural shunt; no intraprocedural complications were observed (fig.C). The 1 month follow–up CTD was negative for RLS. Conclusions Residual ASD is a rare late complication after surgical closure of the ASD(1). We describe the case of a successful transcatheter closure of a small ASD in the presence of a deficient posterior–inferior rim which, in general, is considered an unfeasible anatomy for percutaneous procedure (2). The appropriate choice of treatment for a residual ASD should be carefully considered on a case–by–case basis. Reference 1) S. Ogura;Y.Takaya1; T. Akagi; K. Nakagawa; H. Ito.Percutaneous closure of residual atrial septal defect after surgical closure. Cardiovascular Intervention and Therapeutics (2021) 36:256–259 2) M. Chessa; G. Butera; A. Giamberti; R. M. Bini; M.Carminati. Transcatheter Closure of Residual Atrial Septal Defects After Surgical Closure. Journal of Interventional Cardiology vol.15, No. 3,2002

Whole-Genome Sequencing Reveals Mutational Signatures Related to Radiation-Induced Sarcomas and DNA-Damage-Repair Pathways

Radiation-induced sarcoma (RIS) is a rare but serious late complication arising from radiotherapy. Despite unfavorable clinical outcomes, the genomic footprints of ionizing radiation in RIS development remain largely unknown. Hence, this study aimed to characterize RIS genomes and the genomic alterations in them. We analyzed whole-genome sequencing in 11 RIS genomes matched with normal genomes to identify somatic alterations potentially associated with RIS development. Furthermore, the abundance of mutations, mutation signatures, and structural variants in RIS were compared with those in radiation-naïve spontaneous sarcomas. The mutation abundance in RIS genomes, including one hypermutated genome, was variable. Cancer-related genes might show different types of genomic alterations. For instance, NF1, NF2, NOTCH1, NOTCH2, PIK3CA, RB1, and TP53 showed singleton somatic mutations; MYC, CDKN2A, RB1, and NF1 showed recurrent copy number alterations; and NF2, ARID1B, and RAD51B showed recurrent structural variations. The genomic footprints of nonhomologous end joining are prevalent at indels of RIS genomes compared with those in spontaneous sarcoma genomes, representing the genomic hallmark of RIS genomes. In addition, frequent chromothripsis was identified along with predisposing germline variants in the DNA-damage–repair pathways in RIS genomes. The characterization of RIS genomes on a whole-genome sequencing scale highlighted that the nonhomologous end joining pathway was associated with tumorigenesis, and it might pave the way for the development of advanced diagnostic and therapeutic strategies for RIS.

Primary Adenocarcinoma at Ileostomy—A Rare Late Complication: Three Cases and Literature Review

Introduction Primary adenocarcinoma at the ileostomy site is a rare complication, with only 57 cases reported in the literature. The primary diagnosis is usually ulcerative colitis (UC) or familial adenomatous polyposis (FAP). Cases We present 3 cases of adenocarcinoma at the ileostomy site. Two of our 3 cases have a previous diagnosis of cancer, with no previous diagnosis of UC or FAP. Discussion Adenocarcinoma at ileostomy is a rare late complication of permanent ileostomy. Possible etiologies are chronic irritation and changes in bacterial flora that lead to metaplasia and cancer development. Conclusion Adenocarcinoma at the ileostomy site is a rare complication with good prognosis if detected early. Diagnosis is by biopsy, and treatment is surgical. A biopsy should be considered in any abnormal growth around the ileostomy.

Successful pseudo-autologous stem cell transplantation for donor-derived Burkitt lymphoma occurring 9 years after allogeneic transplantation.

Donor-derived hematological malignancies have been recognized as rare but serious late complications in allogeneic hematopoietic stem cell transplantation (allo-HSCT) recipients. Most cases in the literature were diagnosed as myelodysplastic syndrome or acute leukemia, with very few malignant lymphoma reported. We herein present another case of donor-derived Burkitt lymphoma that occurred 9 years after allo-HSCT under continued administration of immunosuppressants for chronic graft-versus-host disease (GVHD). The patient achieved a partial response after rituximab-combined intensive chemotherapy. To reduce the risk of relapse and to avoid organ toxicities due to repeated chemotherapies, we performed upfront high-dose chemotherapy followed by stem cell rescue using donor-derived CD34+ cells, called pseudo-autologous HSCT (pASCT), and adjusted immunosuppressants appropriately. The patient remained disease-free for 23months after pASCT without exacerbation of cGVHD. Although the observation period has been relatively short and longer follow-up is needed, pASCT may be a feasible option for donor-derived lymphoma even in patients with active cGVHD.

Stenosis of pancreaticogastrostomy after pancreaticoduodenectomy (a case report)

Formation of the pancreaticodigestive anastomosis after pancreaticoduodenectomy (PD) is the “Achilles’ heel” of this operation. Early postoperative complications are well known and well described in the scientific literature, but there are few works on late postoperative complications. Stenosis of pancreaticodigestive anastomoses are rarely mentioned, and the vast majority of publications are focused on stenosis of pancreaticojejunostomy. Regarding stenosis of pancreaticogastrostomy, there are only a few publications of clinical cases in the English-language literature. The aim of the work was to analyze the case of stenosis of pancreaticogastrostomy after PD. Results. The article presents a clinical case of pancreaticogastrostomy stenosis in a patient 5.5 months after PD. The study included the analysis of inpatient and outpatient charts, data of clinical, laboratory and instrumental examinations. Features of the pancreaticogastrostomy formation in clinic are listed, complaints of the patient during the development of stenosis are revealed, results of additional methods of diagnostics and also operative treatment – neopancreaticogastrostomy that was carried out are described in detail. Conclusions. Stenosis of pancreaticogastrostomy is a rare and little-studied late complication of pancreaticoduodenectomy. The clinical course of the pancreaticogastrostomy stenosis, the results of laboratory and instrumental methods did not allow to state with certainty the diagnosis of acute pancreatitis. The formation of neopancreaticogastrostomy is the operation of choice for the described pathology treatment.

Pulp hyperemia: analysis of treatment based on the results of a survey of dentists

The purpose of the study: to analyze the methods of treatment of deep caries by dentists of the Samara region.Materials and methods. In 2021, 220 specialists of the Samara region were surveyed on the treatment of deep dental caries on the bases of budgetary and extra-budgetary organizations. The questionnaires contained 20 questions that provided information about the level of professional training of specialists, the degree of their awareness of quality criteria and standards, the use of modern technologies and the implementation of each stage of deep caries treatment. The statistical significance of the obtained results (p) was calculated using the Student’s criterion (t) and its interpretation based on the standard table of critical values of the Student’s coefficient. Spearman’s rank correlation method was used to determine the strength and direction of the correlation.Results. The majority of dentists – 89.9% use a medical pad in the treatment of deep caries, of which 38.7% of respondents use this pad only when accidentally opening the tooth cavity. Based on the answers to the questionnaire, 77.0% of respondents prefer calcium-containing self-hardening medical pads and only 4.6% use MTA-based pads. A comparative analysis of doctors with rare early complications and doctors with frequent early complications showed that doctors of the second group apply a medical pad when opening the tooth cavity 25% more often than doctors of the first group when performing necrectomy in full volume in both groups. A comparative analysis of doctors with rare late complications and frequent late complications did not reveal a statistical significance between the imposition of a medical pad and the opening of the tooth cavity during a complete necrectomy in doctors of the first and second groups. Complications after the treatment of deep caries may be due to an incorrect interpretation of reversible and irreversible changes in the pulp, and in this regard – incorrectly chosen treatment tactics.Conclusion. The success of the treatment of deep carisea largely depends on the correct determination of reversible and irreversible changes in the pulp of the tooth.

Prandial Diarrhea: A Rare Late Complication After Percutaneous Endoscopic Gastrostomy With Jejunal Extension Tube Placement

Prandial Diarrhea: A Rare Late Complication After Percutaneous Endoscopic Gastrostomy With Jejunal Extension Tube Placement

Brachial artery aneurysm as a late complication of arteriovenous fistula.

Brachial artery aneurysm (BAA) is a rare late complication of arteriovenous fistula (AVF). It brings the risk of peripheral embolism and hand ischemia and is defined by brachial artery diameter above 10 mm or by regional dilatation by >50%. BAA is described in the literature in closed radiocephalic arteriovenous fistulas after kidney transplantation. The aim of the study was to analyze the prevalence of BAA and of their more dangerous forms. A observational one center study performed on patients after kidney transplantation with AVF or arteriovenous graft (AVG). We invited all patients followed up for kidney transplantation in our center. Arterial diameter greater than 10 mm was considered as a brachial artery aneurysm to simplify the detection and evaluation of aneurysms. About 162 patients with AVF after kidney transplantation were examined between 4/2018 and 4/2020. Brachial artery aneurysm was detected in 34 patients (21%) with AVF or AVG, of them 7 had confirmed wall thrombi. AVF flow volume of more than 1500 ml/min increased the risk of BAA development by 4.54x. Eight aneurysms were treated surgically. After this surgery, the primary patency was 87.5% in 12 months. Brachial artery aneurysm was relatively frequent in our study compare to the literature. Aneurysm or dilatation of the brachial artery is more frequent in functional AVFs. Surgical correction is necessary in cases of complicated aneurysms to prevent distal embolization.

Bone Graft Erosion into Hypopharynx Presenting with Stridor: Rare Late Complication Following Cervical Spine Surgery.

Cervical bone graft erosion occurs in about 2% of patients as a late complication following cervical spine fusion surgery. The usual presentation in such patients is dysphagia. Stridor is a very rare late complication of such bone erosions. We report a 66-year-old gentleman who presented with stridor, describe the management thereof.