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A rare cause of hematuria; a case report of sarcoidosis
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Yaşarİncekara1
ErdoğanÇetinkaya2
İsmailUlus3
MustafaÇörtük1
EfsunGoncaUğurChouseın2
Umutİlhan2
RamazanEren2
1Faculty of Medicine, Department of Chest DiseasesKaramanoğlu Mehmetbey UniversityKaramanTurkey
2Yedikule Chest Diseases and Thoracic Surgery Training and Research HospitalUniversity of Health SciencesIstanbulTurkey
3Urology DepartmentBagcilar Training and Research HospitalİstanbulTurkey
Yaşar İncekara1, Erdoğan Çetinkaya2, İsmail Ulus3,Mustafa Çörtük, Efsun Gonca Uğur Chouseın2, Umut İlhan2, Ramazan Eren2
1-)Karamanoğlu Mehmetbey University Faculty of Medicine, Department of Chest Diseases, Karaman,Turkey
2-) Yedikule Chest Diseases and Thoracic Surgery Training and Research Hospital, University of Health Sciences,, Istanbul, Turkey
3-) Urology Department, Bagcilar Training and Research Hospital,İstanbul Turkey
Abstract
Background
Sarcoidosis is a chronic multisystem granulomatous disease most frequently involving the lungs and hilar lymph nodes. Genitourinary involvement, especially bladder sarcoidosis, is extremely rare.
Case Presentation:
We report a 52-year-old female with a history of pulmonary sarcoidosis who later developed hematuria. Initial thoracic imaging and CP-EBUS revealed non-caseating granulomatous inflammation, consistent with sarcoidosis. After six years of follow-up, the patient developed urinary symptoms. Cystoscopy revealed raised bladder lesions, and biopsy confirmed chronic non-caseating granulomatous inflammation. The patient was treated with corticosteroids and later azathioprine was added due to persistent disease activity.
Conclusion
Bladder involvement in sarcoidosis is rare and often difficult to diagnose due to nonspecific findings. Careful monitoring of sarcoidosis patients, including assessment for urinary symptoms, is essential for early detection and timely management.
Keywords:
Sarcoidosis
Bladder
Hematuria
Case report
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Introduction
Sarcoidosis is a chronic multisystem granulomatous disease of unknown cause. The most commonly affected organs are the hilar lymph nodes (LN) and lungs (about 90% of all cases). The typical morphological finding is multiple non-caseating granulomas formed by fibroblasts, lymphocytes, and macrophages (1). The clinical manifestations of sarcoidosis vary considerably and depend on the affected organ, the number of organs involved, the severity of granulomatous infiltration, and whether fibrosis has developed along with functional impairment. In general, corticosteroids are the first-line treatment for patients requiring therapy. Although corticosteroids can maintain disease control in most sarcoidosis cases requiring long-term maintenance therapy, relapses may still occur. In patients who do not respond to corticosteroids, are corticosteroid-resistant, or cannot tolerate corticosteroid tapering, second-line agents such as antimalarials or immunosuppressants can be used as alternatives (2).
This case report presents a patient initially without urological symptoms who, during follow-up, was diagnosed with bladder sarcoidosis, a rare form of involvement.
Case Report
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A 52-year-old female patient presented to the clinic with complaints of fatigue and cough lasting for three months. A chest X-ray at the referring center revealed bilateral hilar fullness (Fig. 1), prompting referral for further investigation and treatment. The patient had a history of diabetes mellitus, treated with oral antidiabetic medication, and a history of thyroid surgery (thyroidectomy), requiring levothyroxine. No additional illnesses or medications were reported. Her family history was unremarkable. Physical examination showed normal respiratory sounds, with an oxygen saturation of 97% on room air, and other system examinations were within normal limits. Laboratory tests revealed an elevated serum angiotensin-converting enzyme (ACE) level of 93.9 U/L (reference range: 8–52 U/L), while other blood tests, including calcium, were within normal limits.
Fig. 1
Chest X-ray demonstrating bilateral hilar fullness.
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Thoracic computed tomography (CT) showed bilateral hilar and mediastinal lymph nodes. A convex probe endobronchial ultrasound (CP-EBUS) was performed, revealing multiple conglomerated lymph nodes in the right paratracheal and subcarinal areas, the largest being 25 mm in diameter, with three samples taken from each site. Cytological findings were reported as consistent with non-caseating granulomatous inflammation. The patient was followed up without medication for six years.
In a follow-up thoracic CT, a nodule with irregular borders, 1 cm in diameter, was observed in the medial segment of the right middle lobe, along with secondary pleuroparenchymal changes in the basal segments of both lower lobes. A PET-CT scan showed newly detected lymph nodes with moderate to intense hypermetabolism in supradiaphragmatic and infradiaphragmatic lymph node stations, consistent with granulomatous disease. Splenic hypermetabolism was also observed. Given the elevated serum ACE level (140 U/mL), the patient was started on methylprednisolone 20 mg once daily in 2019.
During tapering, the patient developed dark-colored urine and hematuria, leading to referral to the urology clinic. Cystoscopy revealed raised mucosal lesions (Fig. 2), and biopsy confirmed chronic non-caseating granulomatous inflammation. Corticosteroid treatment was continued. Three months later, follow-up cystoscopy showed partial regression of the lesions. Azathioprine 50 mg/day was added after rheumatology consultation, while methylprednisolone was maintained at 4 mg/day. The patient’s follow-up and treatment are ongoing.
Fig. 2
Cystoscopic image showing lesions bulging from the mucosa.
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Discussion
Sarcoidosis is a multisystem inflammatory disease characterized by the formation of non-caseating granulomas. The most common sites of involvement are the lungs and hilar lymph nodes (3). Although many patients remain asymptomatic or require minimal therapy, sarcoidosis can affect almost any organ system. Genitourinary involvement, however, is rare (4).
Bladder sarcoidosis is particularly uncommon and difficult to diagnose, as there are no specific laboratory or radiological markers (5). In the literature, only a limited number of bladder sarcoidosis cases have been reported, often presenting with hematuria or irritative urinary symptoms. Our patient initially had no urological complaints, but later developed hematuria, leading to the diagnosis.
As with other organ involvement, corticosteroids remain the mainstay of treatment. Immunosuppressive agents, such as azathioprine, may be considered in patients with relapsing or refractory disease. Bladder involvement should be carefully monitored, as it can lead to hydronephrosis or, rarely, be associated with metachronous bladder cancers (5).
Due to the lack of diagnostic tools beyond symptoms and histopathology, delayed diagnosis and underreporting of bladder sarcoidosis are possible. Therefore, clinicians should remain vigilant and include genitourinary evaluation in the follow-up of sarcoidosis patients.
Declarations
Conclusion
Bladder sarcoidosis is an extremely rare manifestation of sarcoidosis and should be considered in patients presenting with new urinary symptoms during follow-up. Early recognition and appropriate management may prevent complications and improve outcomes.
Consent to publish
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Written informed consent was obtained from the patient for publication of this case report and any accompanying images.
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Funding
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Ethics Statement
Ethical approval
was waived due to the single-patient case report nature.
Competing of Interest
The authors declare no competing of interest.
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Clinical trial registration
Not applicable.
Electronic Supplementary Material
Below is the link to the electronic supplementary material
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Author Contribution
Conceptualization, Y.İ.; methodology, Y.İ and E.Ç.; software, Y.İ.; validation, Y.İ.,R.E.; formal analysis, Y.İ and E.Ç.; investigation, Y.İ. and E.Ç; resources, Y.İ.; data curation, Y.İ , R.E.; writing—original draft preparation, Y.İ.; writing—review and editing, Y.İ. and M.Ç; visualization, M.Ç; supervision, Y.İ.; project administration, Y.İ. All authors have read and agreed to the published version of the manuscript.
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Data Availability
The datasets generated and/or analysed during the current study are not publicly available due to institutional restrictions but are available from the corresponding author on reasonable request.
References
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-) Kalia V, Vishal K, Gill JS, Gill A. Ureteric sarcoidosis–a rare entity. Br J Radiol. 2010;83(996):e247–8. 10.1259/bjr/17908733. PMID: 21088080; PMCID: PMC3473606.
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-) Block NL, Kava BR. Genitourinary sarcoidosis: An essential review for the practicing clinician. Indian J Urol. 2017 Jan-Mar;33(1):6–12. 10.4103/0970-1591.195724. PMID: 28197023; PMCID: PMC5264196.
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Total words in MS: 963
Total words in Title: 10
Total words in Abstract: 126
Total Keyword count: 4
Total Images in MS: 4
Total Tables in MS: 0
Total Reference count: 5