Juvenile Papillomatosis of the Breast: a Report of Two Cases with Review of the literature.

Background: Juvenile papillomatosis (JP) of the breast is a rare and benign proliferative disorder affecting young women. The affected patients tend to have an increased risk of breast cancer development during follow-up. Objective: This article aims to highlight a rare entity of breast disease, that harbor risk of breast cancer. Case Presentation: Here, we present 2 cases of JP in young females; the first case is a 13 year-old presented with spontaneous nipple discharge, while the other patient is a 24 year-old presented with a right breast lump. Both patients had a total excision of the breast lesions, revealing JP at histology. Discussion: Juvenile Papillomatosis is considered a clinicopathological entity and is usually misdiagnosed as fibroadenoma clinically and radiologically, which requires histological correlation. The histologic findings are well-defined (hyperplasia, papillomatosis, and multiple cysts with foamy histiocytes).The controversy in management between surgery and observation is because of insufficient knowledge about the direct relationship between JP and subsequent cancer. Conclusion: Considering the risk of developing breast cancer in JP, enrolling patients and their families in a close follow-up and surveillance program is crucial.

Swiss cheese disease: A rare case of juvenile papillomatosis of the breast mimicking carcinoma in a young female.

Juvenile papillomatosis is a rare benign proliferative lesion of breast seen in young females. These patients have been reported to have a strong family history for carcinoma breast. It is rarely diagnosed preoperatively but has distinct histopathological features on postoperative examination. The subsequent treatment and prognosis vary accordingly. Here, we report a case of a 38-year-old female who was operated with a preliminary diagnosis of Mucinous carcinoma of left breast.

Biliary papillomatosis.

Biliary papillomatosis (BP) is a rare disorder of the biliary tract characterized by the presence of multiple papillary adenomas spread along the biliary tree. Although benign, it carries a significant risk of malignant transformation. Due to low sensitivity and specificity of conventional radiologic modalities, the diagnosis as well as estimation of disease extent is difficult. Endoscopic ultrasound (EUS) and endoscopic retrograde cholangiopancreaticography (ERCP) are superior although direct peroral cholangioscopy (POC) is currently the most accurate diagnostic method. Mainly because it provides more detailed information and makes targeted histological diagnosis possible. The treatment of biliary papillomatosis consists of surgical resection, liver transplantation (LT) or a combination of both. Unfortunately, the recurrence rate after radical surgery without LT remains high due to the diffuse distribution of the disease.

A rare case: Endobronchial solitary mixed papilloma.

Endobronchial solitary papillomas are extremely rare lung neoplasms originating from the bronchial surface epithelium. They often present with cough or recurrent hemoptysis. These tumors are benign, but they should be followed closely because they may even have a low probability of malignant transformation features. It should be kept in mind that malignancy may develop especially if the patient is a smoker. Although the etiology is not known for certain, it is thought to be caused by human papillomavirus in some cases. A 43-year-old male patient was admitted with a complaint of chronic cough. Rigid bronchoscopy was performed for diagnostic and therapeutic purposes after imaging techniques revealed a lesion obstructing the lumen of the right main bronchus. The pathology result was reported as mixed bronchial papilloma. We aimed to present our case because of its rarity and to indicate that chronic cough must be further evaluated.

Recurrent laryngeal papillomatosis: multimodal therapeutic strategies. Literature review and multicentre retrospective study.

Objectives: Recurrent respiratory papillomatosis (RRP) is a benign, rare disease caused by Human Papilloma Virus (HPV) that can be divided into juvenile and adult forms. The course of the disease is variable, but is usually more aggressive in the juvenile form. The standard surgical treatment is represented by CO2 laser resection, although photoangiolytic lasers represent a valid alternative. Adjuvant therapies have been proposed for disease control in case of frequent surgical resections or spreading into the lower airways. In recent years, the development of immunotherapy led to the use of bevacizumab either intratumorally or intravenously, but the most promising therapeutic development is represented by HPV vaccination. This paper aims to present a narrative review of the literature and the experience of three different University Centres in the treatment of RRP. Methods: A retrospective analysis of the clinical charts of all patients affected by laryngeal papillomatosis and treated in three different University Centres between 2002 and 2022 was performed. The following parameters were collected: sex, age at first evaluation, sites of larynx involved, HPV type, type of first surgical treatment, presence and number of recurrences, surgical treatment of recurrences, adjuvant therapies, side effects and status at last follow-up. Results: Seventy-eight patients were available for evaluation. Of these, 88% had adult onset RRP (Ao-RRP) and 12% juvenile onset RRP (Jo-RRP). The glottis was the most frequently involved subsite; all patients were submitted to surgical resection with CO2 laser under general anaesthesia. Recurrences appeared in 79% of the patients, the patients who did not recur were all adults. The mean number of recurrences was 9 (range 1-110). Recurrences were more frequent in children (M = 20; range 2-110) than adults (M = 5; range 1-21). Thirty-two (52%) of the 62 patients who recurred were re-treated with CO2 laser under general anaesthesia, while office-based treatment with a photoangiolytic laser was preferred in the remaining 30 (48%) patients. Adjuvant treatments were applied in 26 patients. The analysis of the course of the disease showed that in the 9 patients with Jo-RRP, 6 (67%) were free of lesions at the last follow-up, while the other 3 (33%) had papillomas. Of the 69 patients with Ao-RRP, 53 (77%) were alive and free of disease at the last visit, 14 (21%) were alive with disease, 1 (1%) was lost at follow-up and 1 (1%) died for other disease. Severe side effects were not observed except for 2 patients, who developed posterior glottic stenosis. Conclusions: Our results confirmed the literature review. RRP is a potentially aggressive disease, especially in juvenile onset. Surgical resection is still first-line treatment, but in case of multiple recurrences the use of adjuvant therapies must be taken into consideration.

Posterior fossa choroidplexus papilloma in the pediatric population: case series and literature review.

Choroid plexus papillomas (CPPs) are rare benign neoplasms which are particularly uncommon in the posterior fossa in children. We herein present a case series of five patients treated at a tertiary care hospital. A comprehensive literature review was also carried out. The patients treated at the tertiary care hospital were aged between 4 and 16 years. Gross total resection (GTR) was initially achieved in two patients. All patients showed clinical improvement. Moreover, 27 articles published between 1975 and 2021 were selected for the literature review, totaling 46 patients; with the 5 patients previously described, the total sample was composed of 51 cases, With a mean age was 8.2 years. The lesions were located either in the fourth ventricle (65.3%) or the cerebellopontine angle (34.7%). Hydrocephalus was present preoperatively in 66.7% of the patients, and a permanent shunt was required in 31.6% of the cases. The GTR procedure was feasible in 64.5%, and 93.8% showed clinical improvement. For CPPs, GTR is the gold standard treatment and should be attempted whenever feasible, especially because the role of the adjuvant treatment remains controversial. Neuromonitoring is a valuable tool to achieve maximal safe resection. Hydrocephalus is common and must be recognized and promptly treated. Most patients will need a permanent shunt. Though there is still controversy on its efficacy, endoscopic third ventriculostomy is a safe procedure, and was the authors' first choice to treat hydrocephalus.

Os papilomas do plexo coroide (PPCs) são neoplasias benignas raras e, na população pediátrica, são particularmente incomuns na fossa posterior. Apresentamos uma série de casos de cinco pacientes atendidos em um hospital terciário. Além disso, foi realizada uma ampla revisão da literatura. Os pacientes atendidos no hospital terciário tinham entre 4 e 16 anos. Ressecção macroscópica total (RMT) foi inicialmente realizada em dois pacientes. Todos os pacientes apresentaram melhora clínica. Além disso, 27 artigos publicados entre 1975 e 2021 foram selecionados para a revisão da literatura, totalizando 46 pacientes. Somados à série de casos atuais, encontramos 51 pacientes, com média de idade de 8,2 anos. As lesões localizavam-se no quarto ventrículo (65,3%) ou no ângulo pontocerebelar (34,7%). Hidrocefalia estava presente no pré-operatório em 66,7% dos pacientes, e derivação ventricular permanente foi necessária em 31,6% dos casos. A RMT foi possível em 64,5%, e 93,8% tiveram melhora clínica. Para os CPPs, a RMT é o tratamento padrão-ouro e deve ser tentado sempre que possível, especialmente porque ainda existem controvérsias quanto ao papel do tratamento adjuvante. A neuromonitorização é uma ferramenta importante para se atingir a máxima ressecção segura. A hidrocefalia é comumente vista nesses pacientes e deve ser identificada e tratada. A maioria dos pacientes irá precisar de uma derivação permanente. Apesar de persistirem controvérsias sobre sua eficácia, a terceiro-ventriculostomia endoscópica foi a primeira escolha para tratar a hidrocefalia na experiência dos autores e é uma opção segura.

Use of extracorporeal membrane oxygenation during resection of recurrent laryngeal papillomatosis: A case report.

RATIONALE: Recurrent laryngeal papillomatosis (RLP) is a common benign tumor in the larynx of children, which is characterized by high recurrence rate and rapid growth, leading to clinical symptoms such as hoarseness and difficulty breathing. Low-temperature plasma radiofrequency ablation (RFA) is the main treatment, but ventilation problems are often encountered during surgery. PATIENT CONCERNS: We report a case of a 2-year-old child with RLP who underwent low-temperature plasma RFA with the assistance of a laryngoscope. However, the surgery had to be temporarily stopped due to ventilation difficulties and difficulty in maintaining blood oxygen saturation during the procedure. DIAGNOSIS: The child was diagnosed with RLP. INTERVENTIONS: The child underwent low-temperature plasma RFA supported by laryngoscopy assisted by ECMO. OUTCOMES: Despite ventilation problems during surgery, the use of ECMO support helped maintain good oxygen saturation in the child and provided a clear surgical field, enabling the tumor to be quickly and cleanly removed. Therefore, the use of ECMO provided critical support during the surgery. LESSONS: This case highlights the importance of airway management during laryngeal papillomatosis surgery. A thorough airway assessment should be performed before anesthesia, and early use of ECMO can reduce harm to the child and ensure the child's safety.

Office-based 532-nm KTP laser as a therapeutic modality for recurrent laryngeal papillomatosis: efficacy and relative factors.

This study aims to investigate the efficacy of office-based potassium-titanyl-phosphate (KTP) 532-nm laser in the management of recurrent laryngeal papillomatosis (RLP) following other treatments. A retrospective assessment was performed on 55 patients in 259 cases of RLP between 2012 and 2019. Derkay scores were obtained for all patients who underwent 532-nm KTP laser procedure (6 W of power with a continuous output mode) prior to treatment and after treatment. Analysis of parameters is based on the distribution characteristics of data. An ordinal logistic regression was also performed. Patients received a median of 3 (range 1-24) office-based KTP laser treatments. Among them, 96.36% (53 patients) were previously on cold steel equipment, CO2 laser, or microdebrider treatment under general anesthesia, and all previous treatments on them had failed. One patient progressed to invasive cancer, so he was excluded from the following analyses. After final KTP treatment, 36 patients (66.67%) received complete resolution with follow-up time ranging from 12.9 to 80.53 months (median 55.54 months). Results of subjective voice-quality indicators such as VHI-30 and GRBAS all improved greatly at the last follow-up. The initial Derkay scores and treatment intervals were found to be predictive of complete lesion remission. Arytenoid involvement may also correlate with lesion resolution. Serial office-based KTP treatment is an effective option for RLP patients, with ideal disease control and voice quality preservation. KTP laser therapy should be repeated with an interval of 1 month from the beginning of treatment until the lesion has been evaluated and subsided. Non-bulk or scattered laryngeal papilloma is an appropriate indication for KTP laser treatment.

Malignant transformation of a solitary tracheal papilloma in the absence of human papillomavirus infection in a patient with 48, XYYY syndrome: case report.

The diagnosis and management of tracheobronchial papilloma is challenging due to its rarity, and non-specific presenting symptoms. Small percentage undergoes malignant transformation. Herein, we report an unusual case of tracheal papilloma initially misdiagnosed as chronic obstructive pulmonary disease (COPD) in 36-year-old male with triple Y syndrome. It was successfully treated with local debridement and brachytherapy. To the best of our knowledge, this is the first description of brachytherapy for such a condition.

Clinical features and factors affecting prognosis and partial deterioration of ocular papilloma: a retrospective study of 298 cases.

PURPOSE: This study aimed to analyze the clinicopathological features of ocular papilloma, a common benign tumor, and the risk factors related to its recurrence and partial deterioration. METHODS: We collected and analyzed the clinical information of 298 patients (51.68% males) with mean age of 41.54 ± 21.95 years, in the ophthalmology department of the West China Hospital. Clinical and pathological factors that might be related to papilloma recurrence and partial deterioration were studied. RESULTS: The top three papilloma sites were bulbar conjunctiva, eyelid skin and palpebral conjunctiva. Moreover, 3.59% of lesions presented a malignant transformation, and 16.28% of patients had one or more recurrences after an average follow-up of 4.47 years. The multivariate logistic regression model showed that multiple lesions were a risk factor for recurrence (p = 0.022, OR = 3.088, 95% CI: 1.180-8.079), while cryotherapy decreased the recurrence risk (p = 0.044, OR = 0.364, 95% CI: 0.136-0.972). Additionally, elderly patients and lesions on the corneal limbus or cornea had a higher risk of malignant transformation (p = 0.004 and 0.01, OR = 1.086 and 7.827, 95% CI: 1.027-1.150 and 1.629-37.596, respectively). CONCLUSION: Ocular papilloma usually occurs in middle-aged and young patients, with no significant gender differences. Older patients and lesions on the corneal limbus or cornea are risk factors for partial malignant transformation. Finally, multiple lesions were a risk factor for recurrence, and cryotherapy reduced the recurrence rate.

[Mixed Squamous Cell and Glandular Papilloma in the Peripheral Lung:Report of a Case].

Pulmonary papillary tumors are usually occur in the upper respiratory tract, and solitary papilloma in the peripheral lung are extremely rare. Lung papillomas sometimes show the elevation of tumor marker or F18-fluorodeoxyglucose (FDG) uptake, and are difficult to distinguish from lung carcinoma. Here we report a case of mixed squamous cell and glandular papilloma in the peripheral lung. An 85-years-old man without smoking history had been presented with an 8-mm nodule in right lower lobe in chest computed tomographic (CT) 2 years before. Since the diameter of the nodule increased to 12 mm, and positron emission tomography (PET) revealed an abnormally increased FDG uptake in the mass (SUVmax 4.61). StageIA2 lung cancer (cT1bN0M0) was suspected and wedge resection of the lung to make a definitive diagnosis and for treatment was performed. The definite pathological diagnosis was mixed squamous cell and glandular papilloma.

Bloody nipple discharge due to intraductal papilloma in an adolescent girl.

An early adolescent girl was referred to our breast surgery clinic with multiple right-sided breast masses and several months of unilateral bloody nipple discharge. MRI demonstrated multiple enhancing masses in the right breast with intrinsic hypertensive T1 signal of the ducts extending to the nipple. A biopsy showed partially sclerosed intraductal papillomas without atypia or malignancy. Following extensive counselling with the patient and her family, two palpable breast masses and a single central breast duct responsible for bloody nipple discharge were fully excised. Histopathological analysis showed unique overlapping features of resembling intraductal papilloma, nipple adenoma and fibroadenomas. The patient has had resolution of her bloody nipple discharge and excellent cosmetic outcomes post-surgery. Intraductal papilloma is rare in the adolescent population and the risk of concurrent and future malignancy is not well established. Thus, a tailored approach to the work-up and management of paediatric breast masses is essential.

Surgical outcome of different surgical modalities for adult recurrent respiratory papillomatosis.

BACKGROUND: Surgical debulking remains the mainstay of treatment of adult-onset recurrent respiratory papillomatosis (AO-RRP). But there are no guidelines for decision-making of surgical modality. OBJECTIVES: This retrospective study aims to obtain a better understanding of AO-RRP, and select the best surgical modality for RRP among microdebrider, CO2 laser, and KTP laser. MATERIAL AND METHODS: Medical records of 72 AO-RRP patients were reviewed, with at least a 6-month follow-up. We focused on demographic data, the median treatment intervals (MTI) (Days), the survival curves, and treatment-related complications. RESULTS: Seventy-two AO-RRP and a total of 172 surgical procedures were included, with either the microdebrider (n = 46), CO2 laser (n = 102), or KTP laser (n = 24). The MTI of all patients was 230.0[132.0, 455.0] (median[P25-P75]), with microdebrider group (267.50[152.5, 449.5]), CO2 laser group (247.5[145.5, 474.7]), and KTP laser group (107.5[68.3, 330.5]), and there were no significant differences among three surgical modalities on MTI or survival curves (p > .05). The duration of surgery was 44.9[25.6] in the microdebriders group, 48.4 [29.5] in the CO2 laser group, and 51.3[18.4] in the KTP laser group, but there was no significant difference in the operation duration among the three groups (p > .05). Postoperative glottic webs and scar formations were found in 4.3% of patients in microdebrider group, 16.7% of patients in CO2 laser group, and 8.3% of patients in KTP group. CONCLUSION AND SIGNIFICANCE: Microdebrider surgery for RRP took the shortest time and had the lowest rate of postoperative complications. However, different surgical modalities are equally effective in controlling recurrence after RRP surgery.

Early Identification and Intervention in Malignant Transformation of Respiratory Papillomatosis.

BACKGROUND Human papilloma virus is a ubiquitous and preventable disease with the potential to cause recurrent respiratory papillomatosis. These papillomas affect the mucosal surface of the airways and may lead to airway obstruction. The papillomas require excision when breathing is compromised, and may be fatal if untreated. Rarely, these papillomas progress to cancer. CASE REPORT We report the case of a 21-year-old woman with a history of HPV 11- and 16-positive recurrent laryngeal and respiratory papillomatosis (RRP) since the age of 7 months, requiring multiple local resections in her respiratory tract. Chest CT demonstrated multiple cavitary lesions throughout both lungs with a rapidly growing mass that occupied most of her right lung. Imaging supported a diagnosis of malignant transformation to squamous cell carcinoma of the lung. Bilateral involvement of the lungs indicated stage IVa squamous cell lung cancer, which is not curable. CONCLUSIONS Clinicians should suspect malignant transformation in patients with HPV type 11, especially if they have required multiple excisions. Earlier age at onset and number of excisions may be predictors for severity of the disease course. These patients need continued surveillance imaging to allow early interventions if malignant transformation occurs. We present the case of a 21-year-old being diagnosed with an incurable disease that may have been avoided with adequate preventive care.

Application of haemoglobin absorption spectral imaging in laryngeal papilloma surgery.

OBJECTIVE: This study compares the detection rates of haemoglobin absorption spectral imaging and white light imaging in laryngeal papilloma surgery. METHODS: Seventeen patients with laryngeal papilloma who underwent surgery in our department from September 2019 to September 2021 were selected. All patients underwent carbon dioxide laser surgery under a microscope. The lesion sites were explored in white light mode and haemoglobin absorption spectral imaging mode. The pharynx and larynx anatomical sites were evaluated using Derkay's all-position scoring system. The numbers and scores for lesions observed in the two modes were compared. RESULTS: In 17 cases, there were statistically significant differences in the numbers of laryngeal papillomas (Derkay score) detected by white light mode and haemoglobin absorption spectral imaging mode. In 9 of 17 patients (52.94 per cent), the haemoglobin absorption spectral imaging mode showed additional diseased tissues. CONCLUSION: The haemoglobin absorption spectral imaging mode can dynamically identify diseased tissues in carbon dioxide laser surgery under a microscope and improve the laryngeal papilloma detection rate.

Bilateral Vocal Fold Granuloma and Anterior Glottic Web After Papilloma Excision.

We present a case of delayed vocal process granulomas and anterior glottic web after treatment of vocal fold papillomas with a laryngeal microdebrider and CO2 laser ablation. This case highlights a risk of vocal fold granuloma in the setting of recurrent respiratory papillomatosis and microdebrider use, warranting close follow-up and consideration of prophylactic voice therapy, anti-reflux medications, and steroid injection in cases of violation of vocal cord epithelium.