Ludwig’s angina in a child: a case report and literature review

Abstract Background Ludwig’s angina is a diffuse cellulitis in the sub-mandibular space, which extends to the sublingual space. It is an emergency that often occurs in adults as a complication of oral infections. It is rare in children and is particularly life-threatening due to the smaller size, as well as the characteristics in these spaces in a child. This is the case of Ludwig’s angina in a 2-year-old boy, with rapid onset of signs of respiratory discomfort, no dental or systemic etiology, and great evolution. Case presentation A little boy was brought by his mom to the emergency room for the onset a firm swelling in the sub-mental region along with pain and fever, which appeared 3 days prior to the consultation. He was first examined by a pediatrician who prescribed oral broad-spectrum antibiotics (amoxicillin). The symptoms worsened over 48 h, as the little boy presented respiratory discomfort in supine position. He was admitted in the emergency department. Without dysphagia or respiratory distress. The clinical examination showed swelling in the sub-mental and sub-mandibular region with/without trismus or signs of oral infection. The laboratory investigations showed hyper-leukocytosis with a microcytic hypochromic anemia.CRP = 300; HIV test was negative. The computed tomography (CT scan) showed a diffuse abscess in the sub-mental and sub-mandibular and sub-lingual regions.No mediastinal collection was found. The diagnosis of Ludwig’s angina was established. The patient underwent percutaneous surgical drainage of 15 ml of pus, which alleviated his symptoms, the treatment was carried out through broad-spectrum antibiotics, analgesics, and daily cleaning of the wound and change of surgical dressing. Bacteriological exam found gram-positive cocci in chains. The culture showed a Staphylococcus aureus . The patient presented clinical and biological improvement and was discharged after 7 days. Six months follow-up showed a healthy child, without signs of infection or any other complication. Conclusion Ludwig’s angina in children -however rare- is a potentially life-threatening, rapidly spreading, bilateral swelling of the sub-mandibular. Its management is based on airway control, drainage of the collection and broad-spectrum intravenous antibiotics, as well as surveillance of the biological parameters. Early diagnosis and appropriate management enhances outcome and decreases mortality significantly.

Necrotizing descending mediastinitis by Acinetobacter Baumannii in an out-patient: a case report

Abstract Background Complicated cervico-facial cellulitis is an infectious disease which can have fatal prognosis. Necrotizing descending mediastinitis is a rare and fatal complication of cervico-thoracic cellulitis. This is the case of necrotizing descending mediastinitis complicating by a multi-resistant Acinetobacter Baumannii in a 27-year-old female, with early management of hemodynamic, respiratory and surgical emergencies, and very careful post-operative care, resulting in very satisfying outcome. Case presentation The patient presented was first treated with broad spectrum antibiotics for Ludwig’s angina for 10 days, then presented with a swelling of sub-mandibular, sub-mental, and cervical regions, a mild respiratory distress, with clinical enhancement of dyspnea in proclive position, tachycardia of 100 beats per minute, and arterial blood pressure of 10/5. Clinical exam showed a tight trismus, with oral opening inferior to 1 cm, no inflammatory signs in facial and cervical swollen areas, and a saturation of 95% in proclive position. Auscultation suspected a pericardial effusion. CT scan with and without injected contrast medium showed diffuse abscesses of sub-mental, sub-mandibular, retro-pharyngeal, para-pharyngeal regions, along with mediastinal abscesses and pericardial effusion. Trans-thoracic ultrasound showed 2 cm pericardial effusion, preserved function of myocardium, and without signs of tamponade. The diagnosis of necrotizing descending mediastinitis with pericardial effusion was established. The patient underwent a course of wide spectrum antibiotic therapy, low doses of cathecholamine, and a surgical drainage through cervical approach of all implicated zones. The surgical dissection was thorough and difficult due to diffuse fibrosis found in tissues of cervical regions. Two hundred milliliters of pus was evacuated, with a placement of surgical drains and Delbet blades. Bacteriological exam found an Acinetobacter Baumannii sensitive to colistin only. The post-operative outcome showed clinical and biological enhancement; however, a residual mediastinal collection appeared in control CT scan after 48 h, which indicated a surgical revision through mediastinoscopy. The post-operative outcome was satisfying with stabilized clinical, biological, and radiological aspects. Conclusions Necrotizing descending mediastinitis is an infectious disease correlated with a very elevated mortality rate, and management is based on airway control, antibiotic therapy, and surgical treatment, as well as the post-operative intensive unit care. Early diagnosis and appropriate management enhances outcome and decreases mortality significantly.

Bilateral antrochoanal polyps: a case report

Abstract Background Antrochoanal or Killian polyp is a rare polyp of the maxillary sinus in adults. It is generally unilateral, implanted on the lateral wall of the maxillary sinus upon the uncinate process. Bilateral cases are very rare; only 14 cases have been cited from 1980 to 2020. Case presentation Our patient presented with a complete bilateral nasal obstruction, this symptom persisted for more than 10 years, with a perceived prolapse in his throat in the last 4 months. Anterior rhinoscopy showed a polyp that took up the totality of his nasal vestibule. Posterior rhinoscopy showed a polyp bulging behind the uvula. The challenge in this case is related to diagnosis decision; conditions with the same clinical presentation are several. Diagnosis was established through the clinical, radiological, and endoscopic aspects, and confirmed by histological findings. Management was obtained through functional endoscopic sinus surgery; no recurrence was noted in the 12-month follow-up, and clinical signs had completely subsided. Conclusion Diagnosis in bilateral forms of Killian polyp is perplexing; however, its management is generally simple, with excellent outcome. We report the case of the 15th patient along with a systematic literature review discussing bilateral Killian polyps, illustrated by a rare case of enormous bilateral antrochoanal polyps.

Plasmacytoma of the larynx: case report and literature review of laryngeal extra-medullary plasmacytoma

Abstract Background Monoclonal plasma cell neoplasms are rare; laryngeal solitary plasmacytoma is very rare. Case presentation This is the case of a 46-year-old man who has been treated for chronic laryngitis for 8 months; he presented first with dysphonia, dyspnea, and dysphagia. The initial tests showed normal erythrocyte counts, elevated erythrocyte sedimentation rates, elevated CRP, and a thrombocytopenia at 40.000. The rest of the test panel was without incidence. Whole body MRI was performed which showed no additional lesion besides the laryngeal location. Bone marrow biopsy was normal. The patient underwent pan-endoscopy under general anesthesia. The biopsy result showed plasmacytoma. The patient had radiotherapy, after which the patient needed tracheotomy with a good outcome. This work presents a very rare case of laryngeal solitary plasmacytoma, with a literature review of differential diagnosis, as well as current trends in diagnosis, and therapy. Conclusion The essential take-home message is that however, this is a rare entity, it should be considered and treated as a malignant disease that could progress to diseases with worse diagnoses. Timely diagnosis and treatment can allow good outcomes and prevention of progression.

A large and invasive pleomorphic adenoma of nasal cavity: a rare case report and review of literatures

Abstract Background Pleomorphic adenomas are common tumors of major salivary glands. However, the localization in nasal cavity originating from the lateral wall is exceptional. This work is a presentation of a very rare case that presents the diagnostic challenge, considering the multitude of malignant and benign differential diagnosis. A discussion of surgical approach, and the possible complications associated with it. Case presentation This is the case of an invasive nasal pleomorphic adenoma in a 48-year-old patient; the patient underwent complete excision through combined left lateral rhinotomy and functional endoscopic sinus surgery. Conclusion Pleomorphic adenoma is a very rare benign tumor of the nasal cavity. Clinical diagnosis is generally difficult regarding the multitude of differential diagnosis. Confirmation is established upon histological aspect. This is a case of a very rare large pleomorphic adenoma of nasal cavity originating from the lateral wall, associated with differential diagnosis, and surgical approaches, as well as perceivable complications. The strength of this work is to point out the importance of complete surgical extirpation and thorough follow-up to prevent recurrences and malignant transformation.

Cystic lymphangiomas of the tongue: 3 rare cases and a literature review of classifications and therapeutic possibilities

Abstract Background Cystic lymphangiomas of the tongue are rare. The treatment consists of complete resection and management is generally interdisciplinary due to vital, functional, and esthetic comorbidities. This work is a report of three rare cases of cystic lymphangioma of the tongue, with curious mode of presentation and good outcome, as well as a literature review of clinical features, classifications, and therapeutic possibilities. Case presentation This work presents three patients. The first one presented with a lymphangioma in the junction between base and body of tongue; the clinical aspect was first similar to hemangioma, and then the diagnosis was re-established after slight enlargement of the mass. The patient was treated with sclerotherapy with great outcome. The second patient presented with a large lymphangioma which presented a clinical resemblance with a tumor of the tongue, the diagnosis was established upon radiological aspect and confirmed upon histological findings. The third patient presented with a macroglossia which appeared 3 years after excision of the cystic hygroma of the neck; the patient was surgically treated with excellent outcome. Conclusion Cystic lymphangiomas are rare hamartomatous lymphatic malformations, and they have a predilection for head and neck but the lingual localization is quite rare. Treatment is complete excision. Management is generally interdisciplinary. The main objective is to prevent alteration to function, to treat esthetic comorbidities, and prevent speech impediment especially in very young children. This is the cases of three young children with a rare lingual cystic lymphangioma, with adapted treatment and satisfactory outcome, with a literature review of clinical presentation, classifications, and therapeutic approaches.

The challenging management of an assident parotid tumor: a case of solitary plexiform neurofibroma of the parotid facial nerve

Acute otitis media in an infant complicated with osteoarthritis of temporomandibular joint and sub-periosteal abscess of the clavicle

Abstract Background Otomastoiditis is a very frequent affection and a current complication of mal-treated benign ear infections in children. However, this a very rare case of the association of two rare complications of otomastoiditis in a newborn. On the one hand, septic arthritis of the temporomandibular joint which is a very rare condition that is difficult to diagnose, and when unrecognized or not treated accordingly, it can resolve in serious infectious complication and or definitive injury to the temporomandibular joint. On the other hand, osteomyelitis of the clavicle is also very rare, and only a few cases have been cited in the literature concerning infants. Case presentation This 46-day-old infant was brought to pediatric emergency consultation for 2 swelling inflammatory bulges, one in the right mastoid and pre-auricular regions, and another in the right basi-cervical area. The infant was hypertrophic febrile, hypotonic, and pale. He had preserved archaic reflexes. Besides, blood test showed an inflammatory syndrome, inflammatory anemia, and no other abnormalities. Upon supplementary computed tomodensitometry exam, the diagnosis of a combination of septic arthritis of the right temporomandibular joint and sub-periosteal abscess of the ipsilateral clavicle in a context of hypotrophy and malnutrition was suspected. A pus sample was obtained for bacteriological evaluation, after which the infant had a course of intravenous associated antibiotics, along with nutritional assessment and management. Surgical drainage of both collections was performed. The 6-month follow-up was satisfactory, without clinical signs of functional impact on temporomandibular joint, or acromioclavicular joint. Conclusion This work stresses the necessity of thorough clinical examination of infants even in cases of benign ear infections, as well as the importance of adapted treatment and follow-up, which could allow early diagnosis, appropriate treatment, or even prevention of severe complications that can be associated with such benign conditions.

A foreign body lodged in the sub-mental space through to the retro-pharyngeal area: a review of anatomical risks and surgical approaches

Abstract Background Foreign body inclusions are rare in head and neck. They are challenging in regard to anatomical risks and surgical approaches. This is the case of a particular foreign body with a curious trajectory, associated with a brief review of anatomical risks and surgical approaches. Case presentation This is the case of a 25-year-old male who has been attacked with a sharp object, which caused an inclusion of a part of the foreign body in the sub-mental and pharyngeal areas. The clinical exam showed a painful bulging in the sub-mental area, with moderately hemorrhagic sputum and difficulty while swallowing. The intra-oral exam showed a foreign body that was located at the right side of the base of the tongue, and which goes backwards and outwards to penetrate retro-pharyngeal mucosa. The patients underwent an angio-CT scan, which showed a curious fine and sharp metallic object, without direct signs of lesions to the jugular vein or carotid artery or any collateral branches. The patient underwent extraction through cervical approach, with satisfying bleeding control. Intra-oral exploration showed a retro-pharyngeal lesion of 2 cm, without particular bleeding. The cervical lesion and retro-pharyngeal lesions were closed. The lesion at the base of the tongue was of 1 cm, palpable but not accessible to sutures, and a naso-pharyngeal tube was inserted. The patient presented very satisfying post-operative outcome, without any complications. Conclusion When dealing with foreign bodies of head and neck, physicians must be precocious and prepared for the risk of bleeding after extraction. Thorough radiological exploration is necessary as long as the patient is stable. Direct and indirect radiological signs allow the clinician to understand the nature and the trajectory of the object, as well as the damage to collateral structures.

Fortuitous discovery of pulmonary tuberculosis in an asymptomatic, SARS-CoV-2 positive, Moroccan patient

The COVID-19 pandemic has created worldwide change in screening, management, and deadlines for the treatment of tuberculosis, among other latent diseases. Safe access to health-care structures, drug interactions, and ongoing changes in treatment guidelines are the main challenges. The first published cohort of 45 patients with both tuberculosis and SARS-CoV-2 infection presented three different scenarios regarding the timing of these diagnoses, both diagnoses being made within the same week in only nine of these patients. Thus far, tuberculosis has not been shown to influence the pathophysiology of SARS-CoV-2 infection. The association between these two diseases may be incidental. Tuberculosis remains the leading lethal infection worldwide. 2 In patients with preexisting pulmonary tuberculosis and COVID-19, management priorities, drug interactions, and optimal frequency of clinical, biological, and radiological check-ups require clarification. We here report an asymptomatic young patient who had both COVID-19 and pulmonary tuberculosis, raising challenging questions regarding diagnostic tools, treatment, and follow-up.

A rare case of oral cavity lipoma in the inferior vestibule: a case report and literature review

Abstract Background Lipomas are ubiquitous tumors. They are mostly present in the trunk as well as the proximal portion of the limbs. However, 15–20% are located in the head and neck regions, and only 4.4% are found in the oral cavity. They normally present as a bulge with signs of compression, without signs of infiltration. Case presentation This is the rare case of a 36-year-old man, who was admitted to ENT consultation for a bulge in the left inferior vestibule of the oral cavity. The bulging was not painful, with no inflammatory signs, no dental mobility, or abnormalities in the vestibular mucosa. No other masses were found in the general clinical exam. The patient underwent a dental panoramic X-ray, which showed a low opacity lesion next to the left inferior vestibular space, without signs of bone invasion. There was no hypoesthesia in the territory of the alveolar nerve was found. The patient underwent a craniofacial MRI which showed a mass which was hypointense on T1 and hyperintense on T2. Fine needle aspiration was not performed. The patient’s pre-operative blood work showed no abnormalities. He underwent surgical excision under general anesthesia. The incision was carried out in the left inferior vestibular space, 1 cm away from the alveolar gingiva; careful dissection of the mucosa allowed the discovery of a large fatty mass, encapsulated, with a nourishing pedicle, without signs of invasion or ulceration in the adjacent structures. Careful dissection and hemostasis were performed, followed with the closure of the mucosa with Vicryl 4.0. The histopathological exam showed mature adipose tissue. The fat contains few small capillaries within thin fibrous strands. A thin fibrous capsule was seen. Clinical follow-up of 2 months showed no complications. The surgical incision healed perfectly, and no bulging was noticed upon palpation. Conclusions This is the rare case of a vestibular lipoma of the oral cavity; the clinical and radiological aspect allowed consideration of several differential diagnoses. The most important take away lesson is related to the very rich variety of differential diagnosis in oral cavity masses. Clinical and radiological aspects allow the clinician to have diagnostic theories. Only histological results allow to establish a definitive diagnosis.

Diagnosis difficulty of histiocytosis in the thyroid region of a child: a rare case report with literature review of differential diagnoses

Abstract Background Isolated histiocytosis of thyroid region is very rare; clinical history, exam, and radiological aspects are non-specific, and etiological reasoning is quite difficult considering the tremendous number of differential diagnoses. Case presentation This is the case of a 6-year-old girl who came to the emergency room with an acute presentation bulging of the anterior and left lateral regions of the neck. The palpation of the mass showed tenderness; there was no sign of inflammation, nor was there any fistula to the anterior border of the sternocleidomastoid muscle. The patient was stable. She did not have any signs of compression. The initial blood showed anemia and inflammatory syndrome. She underwent cervical ultrasound exam that showed a mass at the expense of the left thyroid lobe; the mass extends through the sub-hyoid muscles to the lateral cervical region. A CT scan with and without contrast injection was performed. It showed a heterogenous mass, which seemed centered in the anterior compartment, and from which it extended to the left lateral compartment, as well as the posterior compartment, invading the prevertebral muscles and englobing the carotid and the internal jugular vein. The patient underwent surgical biopsy. A basal cervical incision was made, dissection with the myo-cutaneous plane. Per-operative observation established that the mass breeched the infrahyoid muscles, as well as the sternocleidomastoid muscle. A biopsy was performed without opening the middle line. The pathological exam showed an eosinophilic granulomatosis, associated with Stembergoid cells. The immune-histochemical exam concluded that the lesion is histiocytosis. The patient underwent a cervicothoracic and pelvic CT scan to rule out systemic forms. The diagnosis of isolated histiocytosis of thyroid region was confirmed. The patient underwent hemithyroidectomy, associated with careful dissection of extension of the mass to lateral compartment of the neck. Postoperative exam showed no abnormalities. No dysphonia and no hypocalcemia were observed. The 8-month follow-up showed satisfactory results, no cervical swelling, and no signs of inflammation or compression. Postoperative naso-fibroscopy was normal. Conclusions The most important takeaway message of this work is that methodical approach of neck masses allows to rule out the most aggressive lesions frequently encountered, which allows clinicians to establish thorough diagnosis and management without further delay.

Biologic Agents in the Treatment of Severe Chronic Rhinosinusitis with Nasal Polyps in Adults: A Systematic Review and Meta-Analysis

Introduction: Severe chronic rhinosinusitis with nasal polyps (CRSwNP) is a type 2 inflammatory disease that significantly impairs quality of life. Biologic therapies offer targeted treatment options; however, comparative data remain limited. Methods: A systematic review and Bayesian network meta-analysis of randomized controlled trials (RCTs) was conducted to compare the efficacy and safety of approved biologics in adults with CRSwNP. Primary outcomes included changes in the Nasal Polyp Score (NPS), SNOT-22 score, and the incidence of serious adverse events (SAEs). Surface under the cumulative ranking (SUCRA) values were used to rank treatment performance. Results: Eight RCTs involving 2,564 patients were included. All biologics significantly reduced NPS and SNOT-22 scores compared to placebo. Dupilumab showed the greatest reduction in NPS (mean difference [MD]: -2.4; 95% credible interval [CrI]: -2.4 to -2.4) and SNOT-22 (MD: -19.2; 95% CrI: -19.2 to -19.1), followed by Tezepelumab (NPS MD: -2.1; 95% CrI: -2.1 to -2.0; SNOT-22 MD: -17.0; 95% CrI: -17.1 to -16.9). GR1802 ranked highest in safety, with the lowest odds of SAEs (odds ratio [OR]: 0.06; 95% CrI: 0.001 to 1.0), followed by Dupilumab (OR: 0.58; 95% CrI: -0.06 to 5.5). SUCRA values confirmed Dupilumab (0.807) and Tezepelumab (0.730) as the top performers in terms of efficacy, and GR1802 (0.954) as the safest option. The certainty of the evidence was high, and the risk of bias was low across most studies. Conclusion: Biologic therapies improve symptoms and reduce polyp burden in CRSwNP. Dupilumab and Tezepelumab are the most effective agents, while GR1802 demonstrates the highest safety profile. These findings support the use of individualized treatment based on specific clinical goals. Further head-to-head trials are warranted to better guide therapeutic decision-making.