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V2.N3 The Interventionalist Journal https: theinterventionalists.con CONSERVATIVE TREATMENT OF HUGE BLADDER NEUROFIBROMA : A CASE REPORT A. E. J. Mohd' F. F | Omar' L. I K. S. Christopher A M G Khairul' S. K | Lai? 'Department of Urology | Faculty of Medicine Universiti Putra Malaysia, Seri Kembangan Selangor | Malaysia ?Department of Pathology and Microbiology | Faculty of Medicine and Health Sciences b Universiti Malaysia Sabah Kota Kinabalu I Sabah Malaysia Correspondence author | A. E.J.Mohd Department ofUrology / Faculty of Medicine Universiti Putra Malaysia, Seri Kembangan Selangor Malaysia. Telephone +60195525878 Email | azrul.eimirul@upm.edu.m DOI | https://doi.org/10.32896/tj.v2n3.6-1 Submitted | 07.07.2022 Accepted | 20.09.2022 Published | 30.9.2022 ABSTRACT | Neurofibroma with involvement of the urinary bladder is a rare condition that has been reported in less than 80 cases reported worldwide We present a 22-year-old lady with a known history of childhood neurofibroma type 1 Incidental findings of a huge pelvic mass during laparoscopic surgery for initially thought of a gynae pathology Further investigation after that with Computed Tomography CT 1 scan displayed a bladder mass occupying the abdomen cavity with regards to neurofibroma of the bladder A core Biopsy of the mass was done and confirmed the pathology Clinically patient was asymptomatic with the mass The patient refused surgical intervention Follow-up for 2 years with serial imaging showed no significant progression of the disease. It is important to determine any Sarcomatoid/malignant changes to decide on further management of bladder neurofibroma. Keywords | bladder neurofibroma neurofibromatosis NF1 I . , | , plexiform neurofibroma INTRODUCTION | possible malignancy changes in the future Neurofibroma of bladder are approximately less Inadequate evidence of other treatment modalities than one-third of these cases are in the pediatric such as radiotherapy and chemotherapy able to population In the bladder neurofibromas arise in salvage of this condition have been reported the nervous ganglia of the bladder wall Most However managing by conservative treatment is Neurofibroma of bladder are identified at young considered still good choice as risk of malignant age and no exception in our case Majority of those changes are varied from 5-20% from collection of patient that are symptomatic presented with pain reported case So far We present a massive haematuria lower urinary tract symptoms or plexiform Neurofibroma of bladder managed by obstruction It was different in this case where it conservative management for up to 2 years was incidentally found during gynaecology following patient decision without evidence of surgery and patient was asymptomatic The way significant changes. mass appeared in CT are worrying of malignancy or possibility presence of sarcomatoid changes. 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theinterventionalist. .com for irregular menses Ultrasound abdominal neurofibroma with no intravesical mass. Multiple assessment showed possible right ovarian cyst, 10 flat sessile lesions generalized scattered over cm in the largest diameter suspected of an ovarian bladder Biopsy was performed by an mass. She was scheduled for laparoscopic right interventional radiologist. Core tissue bladder oophorectomy by gynecology team mass was successfully obtained and sent for Intraoperatively a huge pelvic mass possibly histopathology I Plexiform bladder neurofibroma arising from the bladder was found and it was was confirmed without malignant or sarcoma attached to the anterior abdominal wall. However features seen Findings were discussed with reproductive organs were normal in appearance patient and offered for surgery partial cystectomy Urologist was called-in the operating theatre for However patient was not keen for surgery Serial opinion The procedure was abandoned in view of imaging follow-up with CT and ultrasound was uncertainty of the diagnosis and possible dealing done for 2 years that showed no signs of with malignant tissue without proper progression . suggesting a stable disease. There is investigation CT abdomen pelvic was arrange no hydronephrosis on both kidneys with renal post procedure Further history patient had function urine examination showed normal normal urination without any history of lower parameters. urinary tract symptoms S. haematuria, or history of No genetic test was done for the patient On recurrent urinary tract infection examination pelvic mass was palpable, however Flexible cystoscopy revealed bladder irregular no stigmata of neurofibroma were found on mucosal surface suggesting of bladder examination externally 00 11- Figure 1 | An image taken during laparoscopic surgery showed huge pelvic mass occupying anterior part of pelvic extend superiorly near anterior abdominal wall. 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Figure 4 Figure 4 shows histological image of bladder neurofibroma sample taken using CT guided biopsy at the core of the mass. There are spindle cells proliferation in variable cellularity forming nodular pattern in areas. The spindle cells show mild pleomorphism with wavy nuclei and inconspicuous nucleoli Mitosis is not observed No necrosis is noted Mast cells are present Immunohistochemical study showed the spindle cells are focally positive for S100 No sarcomatoid changes were seen DISCUSSION : pathognomonic pathology finding of Neurofibromatosis type 1 (NF1 ) is an autosomal schwannomas. dominant transmitted disease with various clinical In our case we present a young lady with a known manifestations Neurofibromatosis (NF1 ) case childhood NF type 1 presented with irregular affecting approximately 1 in 3000 individuals. It menses without any urinary symptoms. For can be presented with alterations of skin bladder neurofibroma possibility of malignant pigmentation | iris Lisch nodules 3 and multiple transformation or sarcomatoid transformation is a benign neurofibromas usually constitute the must to o look for From the literature, it was found clinical picture. The genitourinary tract is rarely out about 5-10% tumors underwent malignant involved in NF1, and less than 80 cases were transformation during follow-up; and 12-29% for reported in the literature to date [2] non n-genitourinary involvement [4] None of these Bladder neurofibroma typically occur in young occurred in children The risk of malignant patients with neurofibromatosis type 1 degeneration changes increases with advancing Neurofibroma of the bladder presents early in life age [3] and after an operation for benign and occurs more often in the setting of generalized neurofibromas. [4] disease than as isolated visceral Looking at the CT image and intraoperative neurofibromatosis The mean age at diagnosis is laparoscopic findings, the bladder mass was huge reportedly 17 years ( (range C 1 month to 54 years ), with the largest diameter of 15 cm occupying the and the male-to-female ratio is 2.3:1. anterior abdominal wall and pelvic space Clinical presentations can be voiding/storage displacing bowel superiorly and reproductive symptoms b flank pain with urinary incontinence organ posteriorly Possible malignant changes The diagnosis is confirmed by histopathological need to rule out so possible surgical intervention and immunohistochemical examination can be offered Multidisciplinary discussion with Histologically | the tumors are usually of the oncology | intervention radiologist (TR ) and plexiform and diffuse type. Histopathologically | urology team was conducted Core biopsy was they stain positive for protein S100 with obtained with help of IR to give more ideas of immunohistochemica techniques , and this is the diagnosis C " }, { "page": 4, "image_url": "https://nnts.imgix.net/uploadedfiles/rawocr/1513a17a-ded4-48b3-a16c-dedc9b910b58-5.jpeg?auto=compress&expires=1664515013&or=0&s=43083f006385f3162106edce622ef4cc", "size": { "width": 2481, "height": 3508 }, "words": [ { "text": "TIJ2022", "xmin": 220, "ymin": 128, "xmax": 368, "ymax": 157 }, { "text": "V2.N3", "xmin": 382, "ymin": 128, "xmax": 482, "ymax": 156 }, { "text": "The", "xmin": 1787, "ymin": 128, "xmax": 1849, "ymax": 156 }, { "text": "Interventionalist", "xmin": 1861, "ymin": 128, "xmax": 2132, "ymax": 157 }, { "text": "Journal", "xmin": 2144, "ymin": 128, "xmax": 2265, "ymax": 157 }, { "text": "https:", "xmin": 1839, "ymin": 175, "xmax": 1917, "ymax": 201 }, { "text": "/theinterventionalists.con", "xmin": 1921, "ymin": 174, "xmax": 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In this case the was absent of radiological aspects of neurofibromas are mitotic figure C necrotic cell and spindle cell characteristic : especially on MRI and they can proliferation in nodular pattern suggesting benign frequently evoke the diagnosis b which is findings. Immunohistochemical study shows the confirmed by biopsy We believe MRIcould be an spindle cells are focally positive for S100 with excellent tool for non-invasive follow-up [8] as it presence of Mast cells This distinctive clinical. gives more detail in possible sarcomatoid changes histologic and immunohistochemical findings of mass during follow up. No specific guideline proved a definitive diagnosis. [3] in follow up neurofibroma of bladder in our case Treatment of these tumors has included we used regular ultrasound in 6 month and yearly cystectomy , transurethral resection 1 observation F CT to look for any significant change in size and radiotherapy | chemotherapy | and urinary symptoms. diversion Some of patients who presents with symptoms of bladder overactivity were treated CONCLUSION | with botox intravesical iniection after fail Presentation of bladder neurofibroma are rare. In treatment of oral anticholinergic.[6] this case with huge bladder mass with mass effect For malignant transformation or malignant in abdomen are alarming for malignant changes b peripheral nerve sheath tumor (MPNST), surgery however no evidence of it on histopathology is the mainstay of treatment However oncology examination. Surgery treatment was offered treatment such as chemotherapy and radiotherapy however patient preferred to be regular imaging are also offered even the outcomeis still uncertain surveillance. Follow up for 2 years showed no From a meta-analysis that was done by Cai et al significant changes clinically and radiologically A that study all reported case ofMPNST on different extremities was studied and conclude the ACKNOWLEDGEMENT : prognosis are poor with high chance of local We acknowledge those who have been directly or recurrence. Worse prognosis factors mainly indirectly involved in the management of this associated with NF 1 mutation large size C deep to patient throughout his hospitalization and fascia. high grade C metastases | and location ( (trunk discharge and head and neck). [7] Significant morbidity may be associated with STATEMENT OF ETHICS | neurofibroma of the bladder Involvement of the Ethical approval was not required as this is a case bladder is often extensive : necessitating report Informed written consent to participate was cystectomy in approximately one-third of cases provided by all participants. For surgical treatment option that available are mainly pelvic exenteration with urinary diversion CONFLICTS OF INTEREST | with formation of ileal conduit. The authors declare they have no competing For conservative treatment. a study with follow up interests patient for over a mean follow-up of9. .6 years with none of the patients experienced malignant transformation [1] patient was following FUNDING | Our up for years with regular ultrasound kidney, Not ureter applicable. bladder surveillance So far did not show any change with symptoms and radiologically Was DATA AVAILABILITY STATEMENTS : offered for surgical treatment in keeping worried The detailed that support the findings of this case report are available from the corresponding 10 " }, { "page": 5, "image_url": "https://nnts.imgix.net/uploadedfiles/rawocr/1513a17a-ded4-48b3-a16c-dedc9b910b58-6.jpeg?auto=compress&expires=1664515013&or=0&s=b68095f012d1d536bbbfe896ede71a18", "size": { "width": 2481, "height": 3508 }, "words": [ { "text": "TIJ2022", "xmin": 220, "ymin": 128, "xmax": 367, "ymax": 156 }, { "text": "V2.N3", "xmin": 382, "ymin": 128, "xmax": 482, "ymax": 156 }, { "text": 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