Bilateral Submandibular Swelling Research Articles

Bilateral Submandibular Sialolithiasis: A Case Report

Introduction: Lithiasis is the most common salivary disease in adults. Most salivary stones (80% to 95%) produces in the submandibular gland, 5% to 20% in the parotid gland, while the sublingual gland and minor salivary glands account for no more than 2%. Isolated involvement of one gland is usual. Simultaneous lithiasis of both submandibular glands is less frequent. The objective of this study is to report our experience in the treatment of lithiasis of the bilateral submandibular gland. Observation: Mr. K. M, 68 years old, chronic smoking, consulted in emergency November 2023 at the Mohammed VI Oujda University Hospital, for bilateral submandibular swelling. This swelling has been developing for 10 years and increased in size during meals. The current history dates back to 1 month ago with the increase in the right submandibular mass associated with dysphagia. The clinical examination revealed a right and left submandibular swelling, presence of right inflammatory signs: warm mass, painful on palpation, fixed in the deep plane, with expression of pus from the Wharton orifice. Cervical CT scan injected with contrast product was in favor of bilateral sialadenitis on sialolithiasis complicated by submaxillary abscess on the right. Bilateral submandibulectomy with extraction the calculi is done via an external cervical. No post-operative complications were observed and the patient was discharged from the hospital after two days of hospitalization. Discussion: The family history of sialolithiasis suggests the search for a genetic or familial origin of bilateral lithiasis of the submandibular glands, if there is not an obvious or probable etiology. Conclusion: Bilateral submandibular lithiasis is a rare clinical event but one that must be researsh for even in cases of isolated submandibular lithiasis. Extraction of the calculation is made by radical submaxillectomy by cervical externally, and the results are satisfactory.

It Melts Like Snow: Steroid Responsiveness of Immunoglobulin G4-Related Disease

Dear Editor, A 35-year-old male presented with insidious onset gradually progressive bilateral painless submandibular swelling, followed by periorbital swelling [Figure 1] for the past 7 years. He had no constitutional or sicca symptoms. He had no history of addiction or autoimmunity in the family. Systemic examination was unremarkable. On evaluation, he had raised inflammatory markers with a negative autoimmune panel. Magnetic resonance imaging orbit showed bilateral orbital extraconal soft-tissue masses with contrast enhancement. Incisional biopsy from the extraconal mass was suggestive of fibrocollagenous stroma with aggregation of lymphocytes, plasma cells, and histiocytes without any necrosis or granuloma. On further evaluation, submandibular gland biopsy showed chronic sialadenitis with immunoglobulin G4 (IgG4)-positive plasma cell >10/hpf and IgG4: IgG ratio >40% [Figure 2] without any major organ involvement on imaging. The diagnosis of IgG4-related disease was kept. He was started on oral steroid 0.5 mg/kg/day and observed a rapid clinical response within 15 days of steroid therapy. Later, he was started on azathioprine and continued with tapering dose of steroid. He was followed up every month for first 3 months and then every 3 monthly upto 16 months without any signs of recurrence. For a long-standing indolent disease of 7 years, drastic steroid response with resolution of clinical symptoms was quite evident in our case.Figure 1: (a) Pretreatment bilateral upper eyelid swelling, (b) pretreatment bilateral submandibular swelling, (c and d) during glucocorticoid therapy (day 15) resolution of orbital and submandibular swelling, respectivelyFigure 2: Salivary gland with seromucinous acini and ducts embedded in mildly fibrotic stroma with numerous lymphoid follicles with germinal centers (a and b: H and E). Clusters of plasma cells are highlighted with CD138 immunohistochemical stain (c). IgG4 positive cells, >10/hpf (d)IgG4-related disease (IgG4-RD) is a chronic, systemic, fibroinflammatory condition with variable organ manifestation. CD4+ T-cells and B-cells are central to the pathogenesis causing organ damage and fibrosis. Marked expansion of IgG4-secreting plasmablast in the blood is usually seen in active and untreated cases.[1] Key to diagnosis is the classical histopathological hallmarks of lymphoplasmacytic infiltrate, storiform fibrosis, and obliterative phlebitis. IgG4 RD usually has a good response to steroids but a variable relapse rate. Factors associated with lesser treatment response and relapse were well analyzed in a Japanese multicenter study. Multivariate analysis from the study revealed that intermediate steroid initial dose (0.4–0.69 mg/day), slow tapering regimen (<0.4 mg/day), and shorter disease duration were associated with a significantly lower rate of disease relapse.[2] The study of isolated IgG4 ophthalmic disease also echoed similar results regarding steroid response and relapse, with more relapse in those with elevated serum IgG4 levels and a shorter duration of initial steroid treatment (<5 months). Low-dose steroid maintenance should be considered to avoid recurrence in patients with elevated serum IgG4 levels.[3] Later on, rituximab therapy showed promising results in inducing remission[4] and steroid-free long-term maintenance.[5] Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.

The Case | Bilateral submandibular swelling in a dialysis patient

An 80-year-old male patient presented to our hospital with 1-day history of severe diarrhea, vomiting, and chills. He had a history of long-standing type 2 diabetes, arterial hypertension, coronary artery disease status postangioplasty done 6 years ago without any complications, and end-stage renal disease on maintenance hemodialysis since 2017. He had no history of allergies.

IODODERMA AND SIALADENOPATHY AFTER IODINATED CONTRAST MEDIA EXPOSURE

Introduction Iodinated contrast media (ICM) exposure may elicit a variety of rare adverse reactions, including iododerma and iodine-related sialadenopathy (iodide mumps). This patient develops both after ICM exposure. Case Description A 52-year-old AA female with granulomatosis with polyangiitis (GPA) on hemodialysis (HD) presents to the hospital with one-day onset of bilateral submandibular swelling. She had a CT TAP with contrast a day ago for renal transplant evaluation. In the ED, CT neck with contrast is significant for bilateral enlargement and enhancement of the submandibular glands. The next day, she develops bullous lesions on her extremities and face. Physical exam is pertinent for tense bullae on extremities and face and tender submandibular swelling. Skin biopsy is unrevealing. Serum iodine level is 177,360 (normal 52-109) mcg/L. Clinical diagnoses of iododerma and iodide mumps are made, and the patient is started on IV dexamethasone and sent for HD. Sialadenopathy resolves in a week, and skin lesions resolve over the next three weeks. Discussion Iodide mumps and iododerma are rare complications of ICM. The mechanisms are not believed to be immune-mediated. Both reactions are likely secondary to excess accumulation of iodide in the body. Iodine concentration rapidly increases with impaired renal function; 98% of iodine is renally eliminated and the rest via salivary and sweat glands. Both reactions are generally benign and self-limiting; prior cases have utilized corticosteroids and HD, but it is unclear if these improve outcomes. This is a rare case of a patient with GPA on HD who develops two uncommon reactions together after ICM. Iodinated contrast media (ICM) exposure may elicit a variety of rare adverse reactions, including iododerma and iodine-related sialadenopathy (iodide mumps). This patient develops both after ICM exposure. A 52-year-old AA female with granulomatosis with polyangiitis (GPA) on hemodialysis (HD) presents to the hospital with one-day onset of bilateral submandibular swelling. She had a CT TAP with contrast a day ago for renal transplant evaluation. In the ED, CT neck with contrast is significant for bilateral enlargement and enhancement of the submandibular glands. The next day, she develops bullous lesions on her extremities and face. Physical exam is pertinent for tense bullae on extremities and face and tender submandibular swelling. Skin biopsy is unrevealing. Serum iodine level is 177,360 (normal 52-109) mcg/L. Clinical diagnoses of iododerma and iodide mumps are made, and the patient is started on IV dexamethasone and sent for HD. Sialadenopathy resolves in a week, and skin lesions resolve over the next three weeks. Iodide mumps and iododerma are rare complications of ICM. The mechanisms are not believed to be immune-mediated. Both reactions are likely secondary to excess accumulation of iodide in the body. Iodine concentration rapidly increases with impaired renal function; 98% of iodine is renally eliminated and the rest via salivary and sweat glands. Both reactions are generally benign and self-limiting; prior cases have utilized corticosteroids and HD, but it is unclear if these improve outcomes. This is a rare case of a patient with GPA on HD who develops two uncommon reactions together after ICM.

Individuals’ quality of life in periodontal maintenance therapy - critical literature review.

Cervical necrotizing fasciitis (NF) is a soft tissue infection with a low incidence, characterized by rapid progression and high morbidity and mortality. The purpose of this report is to communicate the case of a patient diagnosed with cervical NF and its successful management. A 54-year-old male consulted after suffering from the condition for seven days. It was characterized by bilateral submandibular swelling, accompanied by fever, dysphagia, odynophagia, which were severely affecting the patient’s general health. Physical examination revealed a painful, erythematous cervical swelling. A cervical computed tomography scan was performed, revealing a gaseous collection in the left mucosal pharyngeal space, extending to the glottis, associated with significant deep plane soft tissue emphysema onon the left side of the neck and with possible involvement of the danger space; pertinent lab findings include 19,190/uL leukocytes and 219mg/L CRP. Broad-spectrum antibiotic therapy was initiated with ceftriaxone and clindamycin. Exploratory surgery, lavage and drainage of the collected material were performed. Streptococcus anginosus was isolated by culture. The patient recovered appropriately showing improvement in clinical as well as in inflammatory parameters, being discharged on the ninth day. He is currently receiving periodical checkups in the surgery polyclinic.

Ludwig's Angina

a i A previously healthy 33-year-old man, with a history of type 1 diabetes mellitus, underwent left lower molar extraction. Two days later, he presented to the emergency department with swelling in the left submandibular area and was subsequently treated with meperidine, codeine, and penicillin G. Two days later, after an unsuccessful resolution, he returned with bilateral submandibular swelling and difficulty swallowing and breathing. Vital signs were as follows: heart rate 140 beats/min, respiratory rate 28 beats/min, blood pressure 220/120 mm Hg, and temperature 39°C. After admission, he was taken to the operating room for an emergency tracheostomy and a drainage procedure. Antibiotic therapy was continued with penicillin G and clindamycin, and he made an uneventful recovery.

IgG4-related Mikulicz's Disease: A Report of 3 Cases

We report three cases of hyper-IgG4 disease with synchronous or asynchronous lymphocytic infiltration onset, IgG4 positive plasma cell infiltration, and fibril formation in multiple exocrine glands and extranodal organs. IgG4-related sialadenitis attracting recent attention has yet to be clarified as a clinical entity. Case 1, a 61-year-old man, had a submandibular gland sample showing IgG4-positive plasma cell infiltration. Case 2, a 61-year-old man, was diagnosed with IgG4-related Mikulicz's disease confirmed by a sublingual gland sample. Case 3, a 57-year-old woman, had a diagnosis of IgG4-related Mikulicz's disease confirmed based on labial and sublingual gland samples. All reported oral dryness and bilateral submandibular swelling. Cases 1 and 2 recovered following Predonine administration tapered from 30 or 20 mg. IgG4-related Mikulicz's disease must be recognized as a clinical entity, together with its diagnostic criteria and treatment. Sublingual gland biopsy should be done to confirm its diagnosis following sublingual gland swelling.

IgG4-related Sclerosing Pachymeningitis

IgG4-related sclerosing disease is a distinctive mass-forming lesion with frequent systemic involvement, most frequently the pancreas, salivary glands, and lacrimal glands. This report describes a case manifesting with a previously unrecognized form of central nervous system involvement. The 37-year-old man presented with signs and symptoms of spinal cord compression at the thoracic level 9. Magnetic resonance imaging revealed an elongated dural mass extending from the fifth to tenth thoracic vertebra. Laminectomy and excision of the mass revealed dura expanded by a dense lymphoplasmacytic infiltrate accompanied by stromal fibrosis and phlebitis. IgG4+ plasma cells were increased and the proportion of IgG4+/IgG+ plasma cells was 85%. The patient also had a 1-year history of bilateral submandibular swelling due to chronic sialadenitis. Thus, IgG4-related sclerosing pachymeningitis represents a new member of the IgG4-related sclerosing disease family affecting the central nervous system. It seems that at least a proportion of cases described in the literature as idiopathic hypertrophic pachymeningitis belong to this disease, especially as some patients have other clinical manifestations compatible with IgG4-related sclerosing disease, such as cholangitis and orbital pseudotumor.

Submandibular space infection: a potentially lethal infection

The aims of this study were to review the clinical characteristics and management of submandibular space infections and to identify the predisposing factors of life-threatening complications. This was a retrospective study at a tertiary academic center. We retrieved and evaluated the records of all patients admitted to the University of Padua Otolaryngology Clinic at Treviso Regional Hospital with the diagnosis of submandibular space infection for the period 1998-2006. The following variables were reviewed: demographic data, pathogenesis, clinical presentation, associated systemic diseases, bacteriology, imaging studies, medical and surgical treatment, and complications. A multivariate logistic regression analysis was undertaken using a forward stepwise technique. Multivariate analysis identified four risk factors for complications. Anterior visceral space involvement (odds ratio (OR) 54.44; 95% confidence interval (CI) 5.80-511.22) and diabetes mellitus (OR 17.46; 95% CI 2.10-145.29) were the most important predictive factors in the model. Logistic regression analysis also confirmed other comorbidities (OR 11.66; 95% CI 1.35-100.10) and bilateral submandibular swelling (OR 10.67; 95% CI 2.73-41.75) as independent predictors for life-threatening complications. Airway obstruction and spread of the infection to the mediastinum are the most troublesome complications of submandibular space infections. Therefore, the maintenance of a secure airway is paramount. Patients with cellulitis and small abscesses can respond to antibiotics alone. Surgical drainage should be performed in patients with larger abscesses, Ludwig's angina, anterior visceral space involvement, and in those who do not respond to antibiotic treatment. Moreover, the clinical assessment in patients with comorbidities, especially diabetes mellitus, requires a high level of suspicion for potential life-threatening complications. Early surgical drainage should always be considered in these patients, even in seemingly less critical cases.

自己免疫性膵炎を合併したＩｇＧ４関連慢性硬化性顎下腺炎の１例

A 77-year-old man with a painless and bilateral submandibular swelling was referred to our hospital. He had already been treated with systemic prednisolone for retrobulbar neuritis. Laboratory examinations showed high serum IgG4 concentrations, and a biopsy of the submandibular gland revealed the infiltration of IgG4-positive plasmacytes. Abdominal CT demonstrated tumor formation in the tail of the pancreas. These findings suggested the patient to have Mikulicz's disease associated with localized autoimmune pancreatitis. Treatment with prednisolone improved the swelling of the submandibular gland. As a result, when a paticent is suspected to have Mikulicz's disease, it is important to consider the possibility of systemic IgG4-related plasmacytic disease.

Gas-forming Ludwig’s angina

A 47-year-old man presented to our emergency department because of progressive swelling of the upper midline neck and dysphagia. Physical examination disclosed bilateral submandibular swelling and protrusion of the tongue....

A case of chronic sclerosing sialadenitis of the submandibular gland complicated by autoimmune pancreatitis

Recently, a new clinicopathological entity of IgG4-related sclerosing disease was proposed. It is characterized by extensive IgG4-positive plasma cells as well as CD4 or CD8 positive T lymphocytes infiltration of various organs, and major clinical manifestations are apparent in the pancreas, bile duct, salivary glands, and retroperitoneum in which tissue fibrosis is pathologically induced. We report a case of chronic sclerosing sialadenitis of the submandibular gland complicated by autoimmune pancreatitis. A 77-year-old man consulted our hospital due to the exacerbation of bilateral submandibular swelling. He presented with bilateral enlargement of the submandibular gland and mediastinal lymph nodes, swelling of pancreatic tail, and markedly elevated serum IgG4 concentrations, so was diagnosed as having chronic sclerosing sialadenitis of the submandibular gland complicated by autoimmune pancreatitis. As dense infiltration of IgG4-positive plasma cells was observed in the submandibular gland, chronic sclerosing sialadenitis would be part of IgG4-related sclerosing disease.

Acute bilateral submandibular swelling following surgery in prone position

Acute bilateral submandibular swelling following surgery in prone position

Bilateral synchronous submandibular lumps in a patient with gastric carcinoma

This case report deals with a patient who was readmitted with a bilateral submandibular swelling after having received primary surgery due to gastric adenocarcinoma 6 months before. After bilateral submandibulectomy both glands were diagnosed histopathologically as metastasis of adenocarcinoma. This is the rare case of a submandibular gland metastasis and the first case of a bilateral synchronous submandibular gland metastasis from gastric carcinoma.

Ludwig's angina: a clinical review.

Ludwig's angina is caused by a rapidly expanding cellulitis of the floor of the mouth and is characterized by a brawny induration of the floor and suprahyoid region (bilaterally), with an elevation of the tongue potentially obstructing the airway. In the pre-antibiotic era, Ludwig's angina was frequently fatal; however, antibiotics and aggressive surgical intervention have significantly reduced mortality. We reviewed nine patients with Ludwig's angina between July 1996 and June 2002, all of whom presented with fever, neck swelling, bilateral submandibular swelling and elevation of the tongue. In eight patients (89%) a dental infection appeared to be the underlying cause. High-dosage intravenous antibiotics directed towards the suspected causative microorganisms were given to all of the patients: two were treated successfully with conservative medical management, while seven underwent surgical drainage (a tracheotomy was necessary in one patient). Routine aerobic cultures were done on samples of drained material and the predominant microorganisms were Streptococcus species in two patients; there were none in the other five. Two patients had post-operative complications, but all recovered.