Adenoidectomy Due to Hyperplasia of Adenoids. Treponema Pallidum as a Potential Causative Pathogen

LARYNGOSCOPE(2024)

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摘要
Tonsils are components of Waldeyer's ring and represent an immune barrier against pathogens in the upper respiratory and alimentary tract. Their main purpose is antigen sampling and immune response stimulation. Hyperplasia of this lymphoepithelial tissue is a multifactorial process and might be based on chronic inflammation.1, 2 Persisting hyperplasia could lead to mechanical obstruction or chronic inflammation of the nasopharynx and an adenoidectomy might be indicated.1, 2 Rarely, however, this is indicated in adults. An enlargement of oral lymphatic tissue could be caused by various pathogens, such as viruses (Epstein–Barr virus, Cytomegalovirus) and bacteria (Haemophilus influenzae, Staphylococcus aureus, group A beta-hemolytic streptococci).1, 2 During the last decades, sexually transmitted infections (STIs) have been on the rise. Globally, the incidence of syphilis varies between different countries. In 2020, 7.1 million people were newly diagnosed with syphilis, with higher incidence rates in men who have sex with men.3 Syphilis is known as a great mimicker of various cutaneous and internal diseases. It is caused by the bacterium Treponema pallidum (TP). The disease progresses in three stages and shows specific clinical symptoms in each phase.4 Primary syphilis is usually characterized by a solitary, painless ulcer, occurring at the entry site of the pathogen. In untreated patients, lymphogenic and hematogenic spread of TP after a few weeks leads to secondary syphilis with possible organ involvement (4). Herein, we report the first case of molecularly verified Treponema pallidum in hypertrophic adenoids in a 23-year-old patient suffering from secondary syphilis. In January 2022, a 23-year-old patient who identified himself as a man who has sex with men (MSM) with no history of previous STIs presented himself to the STI outpatient ward due to a non-itchy, pale, macular exanthema on the trunk as well as palmoplantar lesions existing for 3 months. The clinical picture was highly suspicious for syphilis II, and therefore a specific history was taken. The last unprotected sexual intercourse reported was 2 months earlier with a male random acquaintance. According to national and international guidelines, the patient was examined with respect to STIs. Hepatitis A, B, C, and HIV could be serologically excluded. Syphilis serology showed a definite infection [rapid plasma reagin test (RPR) 1:8, treponema pallidum particle agglutination assay (TPPA) >1:1280, treponema membrane protein A (TMPA) reactive, TMPA IgM reactive]. The patient was screened for other STIs on relevant sites (pharyngeal, urethral, and anal). A multiplex PCR for Neisseria gonorrheae, Chlamydia trachomatis, Mycoplasma genitalium, Herpes simplex virus 1/2, Haemophilus ducreyi, Mycoplasma hominis, TP, Trichomonas vaginalis, Ureaplasma parvum, and Ureaplasma urealyticum showed no infection with mentioned pathogens. Further examinations (including chest X-ray, ENT, and ophthalmological clarification) were inconspicuous. The patient received a single-shot treatment with benzathine penicillin 2.4 MIE injected intramuscularly. At the first clinical visit, the patient mentioned hyposmia. Taking a more detailed history, he reported about a recent adenoidectomy. He experienced hyposmia and recurrent swelling of the tonsils within the last 6 months. The extramural otorhinolaryngologist recommends surgical intervention due to impaired nasal breathing on both sides. Hypertrophic adenoids of grade II were identified as the cause of the symptoms, and adenoidectomy was indicated. Due to an additional maxillary sinus cyst and a posterior septal ridge, a FESS, including endoscopic spur removal, was also performed. After the surgical intervention in October 2021, breathing improved, but impaired olfactory sense remained unchanged. Based on the clinical picture and therefore suspicion of neurosyphilis, the patient presented at the Department of Neurology, and a lumbar puncture showed pathological findings (lymphocytic pleocytosis, 11 lymphocytes, 5 erythrocytes, TPPA in Liquor 1:32, lactate 1,5 mmol/L, mild brain-blood barrier). Due to the high probability of neurosyphilis, the patient received penicillin G 24 Mio iE intravenously per day for a total of 14 days. Afterward, the exanthema faded, and the RPR decreased to 1:2 within 7 months. Hyposmia appeared to be largely unchanged, and the patient reported about a slight improvement of smelling after the penicillin doses. Based on the knowledge of the ENT operation, a post-hoc molecular analysis of the original specimen (formalin-fixed and paraffin-embedded (FFPE)) adenectomy tissue was subjected to DNA extraction, (pan-bacterial) 16S rRNA gene PCR amplification, and sequencing. The exact method, sequencing, and strains are presented as supplementary material (Data S1). Notably, no spirochete bacteria could be detected in tissue by using antibodies against TP. We were able to detect TP (0.1% of generated reads mapped to TP next to commensal bacteria), presenting the very first case report of PCR-verified TP as a potential cause of swollen adenoids. Changes in sexual practices and the high rise in STIs lead to oropharyngeal transmission of STIs. Oral transmission of syphilis and its detection in atypical sites are not uncommon and authors have already described this previously. Primary syphilis (chancre) of the oropharynx is possible, as described by Drago et al., who reminded us that extragenital chancre are seldom (5% of cases), but the most frequent extragenital site is the oropharyngeal mucosa.5 Syphilis II is known to present with broadest clinical manifestations and the potential of (internal) organ involvement.4 In total, syphilitic oropharyngeal lesions are most probably underdiagnosed and underestimated by both patients and clinicians. Considering the aspiring syphilis outbreaks throughout the world, the concomitant changes in sexual practices, and therefore an alteration in site of infections, syphilis should be considered even in atypical sites. Especially in risk groups (like men who have sex with men), the authors recommend keeping TP as a potential causative agent for hypertrophic adenoids in mind. To avoid the risk of unnecessary surgical interventions, if suspected, a PCR for TP and serological testing for syphilis should be performed. Data S1. Post-hoc molecular analysis—16S rRNA gene PCR amplification and sequencing. Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.
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hyperplasia,adenoids,adenoidectomy,Treponema pallidum
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