Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 24  |  Issue : 1  |  Page : 23-27

Idiopathic polypoidal scrotal calcinosis leading to delay in diagnosis of testicular tumor


Department of Plastic Surgery, Sanjay Gandhi Post Graduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India

Date of Acceptance07-Jan-2014
Date of Web Publication16-Jun-2014

Correspondence Address:
Ankur Bhatnagar
Department of Plastic Surgery, Sanjay Gandhi Post Graduate Institute of Medical Sciences, Raibareilly Road, Lucknow - 226 017, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1116-5898.134536

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  Abstract 

Idiopathic polypoidal scrotal calcinosis (IPSC) is a rare and benign condition with disputed etiology and it is characterized by multiple calcific nodular deposits in scrotal skin. Here we report a case of a 45-year-old male patient with testicular tumor and 7 years history of scrotal calcinosis is reported. Discussed is the delay in diagnosis of testicular tumor due to IPSC and difficulty in performing fine-needle aspiration cytology in such cases. In our case, no evidence of cystic structure was found around calcified materials. It was indicated that IPSC might be idiopathic. In addition, highlighted the importance of meticulous clinical examination to accurately diagnose the clinical entity and avoid delay in treatment. They are slow growing asymptomatic tumors. Complete excision of the lesion along with the involved scrotal skin with scrotoplasty of the residual scrotal skin is the treatment of choice. Reports of such rare calcified scrotal nodular lesions especially when associated with other malignant conditions need publication and the treatment protocol shared among the surgeons.

Keywords: Calcinosis, fine-needle aspiration cytology, scrotum, testicular tumor


How to cite this article:
Bhatnagar A, Verma V, Purohit V. Idiopathic polypoidal scrotal calcinosis leading to delay in diagnosis of testicular tumor. Niger J Surg Sci 2014;24:23-7

How to cite this URL:
Bhatnagar A, Verma V, Purohit V. Idiopathic polypoidal scrotal calcinosis leading to delay in diagnosis of testicular tumor. Niger J Surg Sci [serial online] 2014 [cited 2021 Nov 27];24:23-7. Available from: https://www.njssjournal.org/text.asp?2014/24/1/23/134536


  Introduction Top


Idiopathic polypoidal scrotal calcinosis (IPSC) is a rare and benign condition defined as the existence of multiple calcified and asymptomatic nodules within scrotal skin, without any metabolic anomaly. [1] The etiology is disputed. Histologically, IPSC is characterized by the presence of calcium deposits of variable sizes within the dermis, often surrounded by a foreign body type granulomatous reaction. [1] A case of a patient with scrotal calcinosis with co-existent testicular tumor is discussed. Delay in the diagnosis of testicular tumor due to scrotal calcinosis and possible difficulty in performing fine-needle aspiration cytology (FNAC) for testicular tumor in IPSC are highlighted. Emphasized is the need of meticulous clinical examination in such cases of multiple pathologies.


  Case report Top


The present case report is about a 45-year-old male presented with 7 years history of progressively increasing multiple scrotal nodules presented to us mainly for cosmetic disfigurement of the scrotum. He had first noticed these nodules about 7 years back. He refused any management as he was explained that these were benign skin lesions. At 2 years back, he also noticed painless enlargement of right testis. The testis and nodules continued to increase in size, both of which were painless and non-tender with no associated constitutional symptoms. However, under the impression that both the lesions were the same he did not take any treatment. At 6 months later, patient consulted a local surgeon, when FNAC of the test was carried out at an outside center. He was diagnosed as having genitourinary tuberculosis (TB) following which he underwent 6 months of anti-tubercular treatment (ATT). There was no resolution of symptoms. At this stage, he consulted our hospital. No records of cytopathology could be obtained from the previous center.

Cutaneous examination revealed multiple, hard, painless, yellowish, subcutaneous nodules of variable sizes beneath the scrotal skin [Figure 1], along with a non-tender enlarged right testis. No separate epididymal enlargement could be detected. Further systemic examination did not reveal any changes. His past medical history, including family history was unremarkable with no history of scrotal trauma. Patient did not have any past history or family history of tubercular treatment. Chest X-ray was normal. Serum and urinary calcium, phosphates and 1,25-dihydroxyvitamin D levels were normal. Urine examination was normal with no evidence of pyurea. Based on the clinical examination, diagnosis of testicular tumor was made.
Figure 1: Scrotum with multiple scrotal nodules

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A repeat FNAC at our center was suggestive of germ cell tumor with associated IPSC. Smears from right testis displayed clusters and sheets of atypical cells on a background of hemorrhage and necrosis. Atypical cells displayed moderate nuclear pleomorphism, irregular nuclear outline, focal prominent nucleoli and microvacuolated cytoplasm. Cytoplasmic periodic acid Schiff positivity was seen. The smears from left testis showed scant cells infiltrate composed of sertoli cells intermixed with spermatogenic cells with no evidence of malignancy in the left testis. The smear from scrotal skin was cellular.

Computed tomography scan showed enlarged right testis with bilateral epindidymal calcification and two 9 × 9 mm lymph nodes in the aortocaval region. Lymph nodes were considered as non-significant. Multiple nodules were seen in the scrotal skin. After metastatic work-up, he underwent right high inguinal orchidectomy in the department of urology, followed by chemotherapy. He refused any scrotoplasty at the time of orchidectomy. Patient underwent scrotoplasty 3 months after completing chemotherapy. Wide excision of the scrotal skin was done and scrotum reconstructed using the residual scrotal skin [Figure 2].
Figure 2: Excised scrotal skin showing calcified subcutaneous nodules

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On scrotal histopathology, sections showed unremarkable epidermis. The sub-epithelial region had multiple areas of calcification in association with collections of histiocytes and multinucleated giant cells in places. Sclerosis and congested blood vessels were also present [Figure 3]. There was no evidence of malignancy. No identifiable epithelial structure on several sections was observed. There has been no recurrence of both the pathologies 6 months after scrotoplasty.
Figure 3: Histology of the lesion showing calcification

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  Discussion Top


IPSC is a sub-group of idiopathic calcific deposits and it occurs mainly in 20-40-year-old men patient and usually manifests during adolescence. Multiple scrotal nodules are observed in most patients. [1] The nodules are hard and yellowish. The condition is benign and usually asymptomatic but could lead to itching or discharge of chalky material. [1] Hence treatment for IPSC is recommended for esthetic reasons only. This may lead to delay in treatment as well as neglect of other lesions in the external genitalia as in our case.

There have been multiple case reports describing in detail the etiology of IPSC.

It is still controversial whether IPSC is idiopathic. The main focus has been on the role of epidermal cysts in the pathogenesis of IPSC. [2] Epidermal cysts were observed by Swinehart and Golitz [2] in three cases of IPSC and some were calcified with partial or total disintegration of the epithelial walls, associated with an inflammatory reaction. [2] Song developed a pathogenic pattern based on histopathological finding, clarified that cysts (epidermal, pilar or hybrid) are formed, implying calcification of the intracystic keratinous content with enlargement of the cyst and a subsequent attenuation of the wall. [1] This triggers a mononuclear cell inflammation or foreign body granuloma with resorption of the cyst walls and of the keratinous material. Finally, only calcified deposits remain. [1] It was considered that histopathological findings varied with the age of the cysts, the oldest lesions no longer containing epithelial cells. In contrast, a study done by King et al. preferred dystrophic calcification of the dartoic muscle to be the initial event in the genesis of IPSC since epidermal cysts seemed not prone to calcification. [3] No evidence of cystic structure was found around calcified material during Hicheri's research. [1] In our case, the nodules were found separated by fibrous connective tissue and surrounding by infiltrated inflammatory cells and a foreign body-type granulomatous reaction, but without any identifiable epithelial structure or fibrous capsule. Thus, it appears that the etiology for IPSC remains debatable. Moreover, etiology does not influence the management of the lesion which is wide excision and scrotoplasty.

Our case is unique in the sense that a malignant condition, i.e., testicular tumor escaped detection during routine clinical examination and investigations due to multiple scrotal nodules. Moreover, since the patient was assured that the scrotal nodules were benign he was late in reporting his symptoms to the physician.

On initial FNAC, the testicular tumor could not be detected. No reports of the patient could be available, but we assume that the reason for starting ATT with testicular swelling could be due to one of the following reasons. First it is possible that inadvertently the FNAC could have been taken from the scrotal nodules rather than the testis and presence of giant cells and calcification within the aspirate may have influenced the diagnosis of Mycobacterium infection. Second it could be possible that FNAC was done from the testis, but through a necrotic region of the tumor; hence presence of caseous necrotic material in the aspirate could have influenced the diagnosis of Mycobacterium infection. Third it could be possible that the patient had a co-existent tubercular epidimo-orchitis, which is a much more common condition as compared to testicular TB. It is essential for the treating physician not to overtly rely on diagnostic findings and clinical correlation is essential. This situation again reinforces the notion that there is no substitute for thorough clinical examination.

Careful examination remains the best non-invasive diagnostic procedure. Although rare among all neoplasms in males, testicular cancer is the most common neoplasm occurring in adult males aged 15-34 and is the third leading cause of death in this age group. [4] Statistics fail, however, to express the impact this disease has on the young parent and wage earners. Correct early diagnosis, careful staging and appropriate management produce remarkable results. Patients with germinal cell testicular cancer currently have the highest cure rate for any solid tumor occurring in males. [4] 3 year tumor-free survival rates over 95% have been reported in patients with early stage disease and higher than 70% for patients with advanced, widespread cancer. [5] The most common initial symptom is usually painless testicular enlargement or a lump. Tenderness, "heaviness", or actual pain may prompt self-examination, or unrelated trauma may draw attention to hemiscrotal asymmetry. The clear definition of intrascrotal structures can be compromised by the presence of a tense hydrocele, or induration with disorders such as torsion, epididymitis and trauma. [5] Along with the above we believe that extensive deformities of the scrotal skin may also mask underlying pathology as seen in our case.

As technology improves, ultrasonography (USG) is increasingly effective in defining scrotal structures and anomalies. If an epididymal mass can be observed clearly distinct from the testis, particularly in the presence of a large hydrocele, unnecessary surgical exploration may be avoided. [5] Hence if the clinical scenario and histopathology does not match it is essential to perform USG of the scrotum. This was not done in our patient before starting ATT.

Markers, if elevated in association with scrotal mass, serum levels of alpha fetoprotein, human chorionic gonadotropin and lactate dehydrogenase (LDH) indicate testis cancer. Alpha fetoprotein and beta HCG are substances produced in normal embryonic tissues during fetal life. They are not normally produced after birth or are produced in minute amounts. With malignant transformation, synthesis returns and serum levels indicate not only the presence, but also the level of cancer activity. Production is, however, not restricted to testicular tumors. [6] LDH should be considered a non-specific indicator of tumor activity, particularly for seminoma. Since marker elevation varies with tumor bulk and type, normal markers do not exclude testis cancer. In fact, elevation may not be observed in 25-40% of patients with retroperitoneal metastasis. [6] Hence markers should again be interpreted in accordance with the clinical scenario of the patient.

Genito urinary TB is the second most common form of extra pulmonary TB. [7] However, genital TB other than tubercular epididymitis is extremely rare. Isolated tubercular orchitis without epididymal involvement is extremely rare. [7] When labeling a patient as a case of testicular orchitis corroborative evidence should be ascertained. Tubercular Epididymitis and associated orchitis is a common form of genito urinary TB in adults therefore, health professionals may tend to attribute testicular swelling to TB infections especially in people coming from endemic areas with a consequent potential delay in the diagnosis of testicular tumors as observed in our case.

To prevent delay in the diagnosis of testicular tumors in patients, we propose the following measures: (1) Patients who develop swelling of the testis should consult a physician as soon as possible for clinical examination; blind antibiotic therapy should be avoided if possible; (2) if clinical examination reveals a hard swelling of the testis and the typical features of acute urinary infection are absent, an ultrasound scan of the scrotum should be performed as soon as possible; (3) in patients with equivocal ultrasound findings, ultrasound-guided, FNAC may allow an early diagnosis of testicular malignancy; (4) education of patients and their caregivers is needed to implement these recommendations. [8]

It is proven that early detection of tumors improves outcome, often with less intensive treatment. The diagnosis of testicular tumors in this group of patients may be delayed because physicians and patients mistakenly attribute testicular swelling to orchitis as indeed happened in this patient. There is no evidence to suggest that FNAC of testicular tumors predisposes to local recurrence or inguinal lymph-node metastasis. [9] Therefore, use of FNAC is essential in patients with testicular swelling in whom findings of clinical and ultrasound examination are equivocal.

Due to one or many reasons discussed earlier, our patient did undergo FNAC which was proven to be fallacious.

The presence of scrotal calcifications, sinus tracts, a non-satisfactory response to conventional antibiotics and findings of pulmonary or extra-pulmonary tuberculous manifestations in the setting of epididymal and testicular involvement, should strongly suggest the diagnosis of tuberculous epididymo-orchitis. None of these were present in our patient hence we kept testicular tumor as our first possibility.

Many general pathologists may have limited experience with testicular tumors because they account for only approximately 1% of human malignancies. [10] Pathologists must be aware of important diagnostic pitfalls in testicular neoplasia that could affect patient management.

Careful characterization of scrotal contents is the most helpful aid in diagnosis. Ease of examination and procedure is facilitated by a warm, quiet, private examination location.

This minimizes cremaster and dartos muscle activity allowing the testicle to descend freely in the scrotum. With the patient supine, each hemiscrotum should be carefully palpated in sequence, usually beginning with the "normal" side. Immobilization and successful palpation should be achieved before doing FNAC. Using thumb and fourth finger, the hemi scrotal contents are isolated in the anterior compartment of the scrotum. By manipulation, using the thumb, index and middle fingers, the complete volume of the testis can be assessed for uniformity of consistency. The presence of a mass or area of asymmetry should immediately alert the clinician to the possibility of malignancy. Benign intratesticular masses are rare, the most common being epidermoid cyst. This is characteristically located on the surface of the testicle and is <3 mm in diameter. [4] The epididymis normally rests on the dorsum and posterior surface of the testicle. Using a similar technique, the epididymal head, body and tail can be readily delineated from the testis. The presence of scrotal skin or intrascrotal abnormalities may make FNAC challenging thereby increasing false reports. We feel that meticulous clinical technique will increase the success rate in scrotal FNAC. This is even more important in cases with the presence of other scrotal lesions.


  Conclusion Top


IPSC is essentially an idiopathic calcific deposit. Surgical excision is the treatment of choice based on the condition and extent of the nodules, though the pathogenesis remains controversial.

The peak incidence for both testicular tumor and IPSC is the same i.e., 2 nd to 3 rd decade any physician dealing with case of IPSC should thoroughly examine the external genitalia. This oversight on the part of the clinician may lead to delay in diagnosis of a malignant condition which if detected early is potentially curable.

Secondly it is also essential to correlate clinical and investigative findings. Testicular or scrotal TB is a very rare condition and hence corroborative evidence should be gathered before confirming such a rare diagnosis. Pathologists must be aware of important diagnostic pitfalls in testicular neoplasia that could affect patient management. The presence of scrotal skin or intrascrotal abnormalities may make FNAC challenging, increasing false reports. Careful characterization of scrotal contents is the most helpful aid in performing FNAC. Ease of examination and FNAC is facilitated by a warm, quiet, private examination location.

In cases where FNAC remains inconclusive or does not match with the clinical findings scrotal USG should be done. In all cases, clinical examination and clinical findings are paramount. Surgical excision for IPSC must be limited to the scrotal skin since calcified nodules are localized within the dermis. Reports of such rare calcified scrotal nodular lesions specially when associated with other malignant conditions need publication and the treatment protocol shared among the surgeons.

 
  References Top

1.Hicheri J, Badri T, Fazaa B, Zermani R, Kourda N, Jilani SB, et al. Scrotal calcinosis: Pathogenesis and case report. Acta Dermatovenerol Alp Panonica Adriat 2005;14:53-6.  Back to cited text no. 1
    
2.Swinehart JM, Golitz LE. Scrotal calcinosis. Dystrophic calcification of epidermoid cysts. Arch Dermatol 1982;118:985-8.  Back to cited text no. 2
    
3.King DT, Brosman S, Hirose FM, Gillespie LM. Idiopathic calcinosis of scrotum. Urology 1979;14:92-4.  Back to cited text no. 3
    
4.Auld RB. Testicular carcinoma. Can Fam Physician 1985;31:1281-4.  Back to cited text no. 4
    
5.Paul J, Krishnamoorthy S, Teresa M, Kumar S. Isolated tuberculous orchitis: A mimicker of testicular malignancy. Indian J Urol 2010;26:284-6.  Back to cited text no. 5
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6.Bansal R, Agrawal V, Mandhani A. Tuberculosis in postchemotherapy residual masses in germ cell tumor of the testis. Indian J Urol 2011;27:274-5.  Back to cited text no. 6
[PUBMED]  Medknow Journal  
7.Viswaroop BS, Kekre N, Gopalakrishnan G. Isolated tuberculous epididymitis: A review of forty cases. J Postgrad Med 2005;51:109-11, 111.  Back to cited text no. 7
[PUBMED]  Medknow Journal  
8.Vaidyanathan S, Hughes PL, Mansour P, Soni BM. Seminoma of testis masquerading as orchitis in an adult with paraplegia: Proposed measures to avoid delay in diagnosing testicular tumours in spinal cord injury patients. ScientificWorldJournal 2008;8:149-56.  Back to cited text no. 8
    
9.Assi A, Patetta R, Fava C, Berti GL, Bacchioni AM, Cozzi L. Fine-needle aspiration of testicular lesions: Report of 17 cases. Diagn Cytopathol 2000;23:388-92.  Back to cited text no. 9
    
10.Ye H, Ulbright TM. Difficult differential diagnoses in testicular pathology. Arch Pathol Lab Med 2012;136:435-46.  Back to cited text no. 10
    


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