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On-line version ISSN 1806-4841
An. Bras. Dermatol. vol.79 no.1 Rio de Janeiro Jan./Feb. 2004
CLINICAL, LABORATORY AND THERAPEUTIC INVESTIGATION
Clinical analysis and anatomopathologic research on patient prepuces referred to postectomy*
Marice Emanuela El AchkarI; Alexandre Bortoli MachadoII; Maurício José PereimaIII; José Caldeira Ferreira BastosIV
in Pediatry, Joana de Gusmão Children's Hospital
IIDermatologist, Senior Member of the SBD
IIIPediatric Surgeon, Joana de Gusmão Children's Hospital
AND OBJECTIVE: The objective of this study is to analyze the histologic
features of the prepuce with phimosis and the incidence of lichen sclerosus
(LS) causing phimosis.
METHODS: Our prospective study included 40 male patients from 05 to 14 years of age, with phimosis referred for circumcision. The patients were distributed into two groups: those with primary phimosis and those with secondary phimosis. The patients were operated at Joana de Gusmão Children's Hospital, and the specimens were examined separately by 3 pathologists. In accordance with their histopathologic features, patients were distributed into three groups: normal histologic findings, lichenoid infiltrate and LS.
RESULTS: The clinical intercurrences most frequently reported by our patients with phimosis were balanoposthitis and urinary tract infection (UTI). Most patients (65%) did not present with histologic alterations of the skin; in 22.5% we found lichenoid infiltrate; and 12.5% had alterations typical of LS. Most cases found belonged to the acquired group, representing 57.5% of the patients studied. All LS cases occurred in the acquired group. The main surgical indication in our casuistry was failure of clinical treatment (45%), followed by balanoposthitis (25%), narrowing of the prepuce (17.5%), UTI (10%) and associated urinary diseases (2.5%).
CONCLUSION: Our study concluded that LS, as the cause of phimosis, showed an incidence of 12.5%.
Key-words: circumcision; phimosis; lichen sclerosus et atrophicus.
The separation of the prepuce from the glans penis begins at around the sixth month of gestation and involves the keratinization of the prepuce and epithelium of the glans. The keratinization begins at opposed ends, i.e. the corona glandis (or, crown of the glans) and distal margin of the prepuce, to which it extends from both sides. Keratinization leads to forming the preputial space separating the skin from the glans.1
At birth, the penis, like the rest of the body, is premature. In the juvenile penis, the development of balanopreputial adherence, resistance of the preputial orifice to retraction, and greater length of the prepuce are normal and considered physiological conditions.2
In 90% of non circumcised boys, the prepuce becomes retractable at roughly five years of age. From this age onward, impossibility of prepuce retraction is called phimosis.4,5
Recently, surgical treatment for phimosis was replaced by the use of topical substances. Some studies6-9 have shown a 67-95% cure rate with the use of medium and high potency topical steroids (0.05% clobetasol or betamethasone). In the latter studies, the fact that anatomopathologic analyses of the treated prepuces were not performed has prevented knowledge from being acquired that could otherwise inform us about the real incidence of the cause of phimosis.
In cases where the topical treatment is not effective and severe narrowing of the prepuce persists, surgical treatment is indicated.10
Circumcision or postectomy, i.e. partial or total removal of the prepuce, has been practiced as a ritual since pre-Christian times.11
There exists little evidence to assert an association between circumcision and good penis hygiene, and its real effectiveness is very controversial.3,12
The reason for the appearance of the phimotic ring remains unknown, with the exception of cases consequent to recurrent inflammatory processes (balanoposthitis); local trauma leading to the appearance of fissures and secondary fibrosis; and dermatological disorders leading to preputial dystrophy, such as lichen sclerosis et atrophicus (LS).13,14
Lichen sclerosis (LS), also known as balanitis xerotica obliterans in men, was first described by Hallopeau in 1887. In 1892, Darier formally described the histologic characteristics of LS. This disorder may affect all age ranges. Reports have ranged from six-month old children to the elderly, with Caucasians making up the majority of patients. LS mainly affects the inguinal area in percentages varying between 85 and 98% of cases. It occurs more in women, in a ratio of five women to every affected male. Ten in every 15% of LS cases occur in children, the majority involving female genitalia.10,15-18
All ages saw various factors reported with their pathogenesis, like human papilloma virus (HPV) and Borrelia burgdorferi, in association with auto-immune diseases (vitiligo, alopecia areata, diabetes mellitus, pernicious anemia and thyroid disease), trauma and genetic factors related to the human leukocytic antigen (HLA).15,16
A few studies19,20 show the LS incidence in pediatric patients with phimosis and were submitted to postectomy to be roughly 15%. However, the data are not precise due to the fact that the tissue removed by circumcision is rarely examined histologically and that the operation is usually curative, with no relapses of the condition.16
The definitive treatment for late-onset LS cases of the prepuce is circumcision. Treatment with local steroids tends to be effective when the inflammatory mechanism is active and tissue damage reversible, thereby including the initial and intermediary histologic conditions of the disease.
Analyze the histologic characteristics of the prepuce when phimosis is present, as well as the incidence of lichen sclerosis as causing phimosis.
The present study is a cross-sectional descriptive survey with prospective data collection.
This study includes 40 pediatric male patients aged between five and 14 years (median 9.5 years), who were diagnosed with phimosis and had surgical indication as treatment. The patients were distributed into two groups based on disease duration: patients with primary phimosis (since birth) and secondary phimosis (or acquired).21
This study obtained a favorable report from the Federal University of Santa Catarina Committee on Ethics in Research Involving Human Beings, project no 086/2001.
By means of the data collection form, the research supervisors provided information referring to the patient-name, age, place of residence-and their disease-onset of the condition, previous treatment and duration, clinical intercurrences, (balanoposthitis, urinary tract infection (UTI), paraphimosis)-as well as whether there was a family history of phimosis. The research supervisors had the patients sign the free consent form and explained the study to them that was to be performed.
All patients were subjected to total postectomy at the Joana Gusmão Children's Hospital (HIJG). The criteria for surgical indication were as follows: 1) failure of clinical treatment for phimosis, 2) associated urinary disorders occurring along with the treatment, 3) cicatricial narrowing of the prepuce-diagnosed by pediatric surgery--, 4) religious reasons, 5) recurrent balanitis, and 6) recurrent UTI. The operation procedure consisted of binding the balanopreputial space, resectioning the fibrotic and/or narrowed preputial ring, hemostasis of the prepuce, performing a mucocutaneous suture with a simple 5-0 catgut wire and reconstituting the balanopreputial groove.
The excised pieces were conserved in a 10% formol solution and sent to the pathologic anatomy laboratory (IDAP - Anatomopathologic and Diagnostic Institute). Each piece was examined separately by three pathologists. In the laboratory, the pieces were fixed in 10% formol, embedded in paraffin blocks, sectioned with a microtome, and prepared as slides stained in Hematoxylin-eosin. Then, the histologic characteristics of the epidermis, dermoepidermal junction and dermis were observed and described without prior knowledge of the clinical data. In accordance with the histopathologic characteristics, the patients were distributed into three groups: normal histologic findings, lichenoid infiltrate and LS. For the histologic diagnosis of LS, the following pathological alterations were considered, in accordance with Lever:22 (1) follicular hyperkeratosis, (2) atrophy of the stratum malpighii with hydropic degeneration of the basal cells, (3) pronounced edema and homogenization of the collagen in the upper dermis, and (4) presence of an inflammatory infiltrate in the dermis.22
The data were typeset by using Microsoft Excelâ 97. Simple and specific descriptions were used as the statistical approach, with corresponding adjacent percentages.
The prevalence of all factors of interest was described. Data were presented in tables and charts.
All patients come from Santa Catarina state: 37 from the middle region of Greater Florianopolis, two from the middle South-Catarinense region and one from the Vale do Itajai (Figure 1).
Forty patients with phimosis aged five to 14 years, participated in the study. All were indicated for surgical treatment after being assessed by a pediatric surgeon. None of the patients was clinically diagnosed with LS. The distribution according to age groups23 is featured in graph 1.
As for the time of phimosis onset, 17 (42.5%) patients with primary (congenital) phimosis and 23 (57.5%) with acquired phimosis were found (Table 1).
The most frequent clinical intercurrences in patients with diagnosed phimosis were: balanoposthitis (30%) and urinary tract infection (10%). There were no reports of paraphimosis. As for family history, among the 40 patients, 20 (50%) confirmed the history (Table 2). In relation to histopathologic findings, the results are presented in table 1. They agreed fully with all of the anatomopathologic expert reports issued by pathologists. Group 1, with no histologic alterations of the skin, consisted of 26 patients (65%); group II, lichenoid infiltrate, included nine patients (22.5%); and group III, LS, five patients (12.5%).
As for prior topical corticosteroid use, 28 (70%) patients used it for a period varying from 15 days to four years (Graph 2).
The results correlating family history of phimosis with the anatomopathologic findings are exhibited in table 2.
The surgical criteria with respect to each age group are presented in table 3.
Forty patients were studied and submitted to postectomy at the Joana de Gusmão Children's Hospital (HIJG) between May 2001 and June 2002.
The majority of patients (92%) came from the Greater Florianopolis region.
The patients who were submitted to postectomy had previously diagnosed phimosis. Given Bloom4 and Piro's5 criteria, referring to phimosis as the impossibility of prepuce retraction from the age of five onward, thereby believing this alteration to be normal up to the age of four, the casuistry spreads from ages five to 14 years. Among these patients, the highest frequency of postectomies was in the seventh year (17.5%). This agrees with the higher rate of phimosis in the six-to-seven year-old age range found in the literature.8
With respect to phimosis type, the patients were distributed into primary or congenital (n=17; 42.5%) and secondary or acquired (n=23; 57.5%) types. Among the causes of acquired phimosis, the literature cites LS Upon considering this pattern closer, the patients analyzed here with primary phimosis had 76% normal histology and 24% of lichenoid infiltrate; no LS was found in the primary phimosis group. In the cases of secondary phimosis: 56% were normal, 22% with lichenoid infiltrate, and 22% with LS; which included all of the LS cases. These results stood in agreement with the work of Meuli et al. in which 90% of patients with LS demonstrated acquired phimosis.20
The clinical intercurrences that patients with phimosis may present are: balanoposthitis, UTI and paraphimosis.
In relation to clinical intercurrences, the cases most frequently found in this study were balanoposthitis (30%) and UTI (10%). There were no cases found of paraphimosis in these patients. These data resemble those in the literature.26
When analyzing family history, LS was found to be reported in mothers and daughters, sisters and even in mono- and dizygotic twins. The HLA complex appeared to control susceptibility to these inflammatory diseases. Still, conflicting studies do exist, with some authors finding a strong association, and others none at all.15,16,17 In the LS cases found in this study, 50% reported a positive family history, against 50% with a negative history.
The majority of these patients (65%) showed normal histology of skin.27,28,29 Clemmensen30 found 46.5%, and Chalmers,19 76% of histology without alteration in pediatric patients with phimosis, who made up the majority of his cases.
In analyzing what causes phimosis, prior studies have shown the incidence of LS as causing phimosis in children. Chalmers et al.19 found an incidence rate of 14%; Kristiansen et al., 15%,26 and Meuli et al.20 demonstrated LS in 10% of cases. In 40 pieces relating to this study and examined histologically, a 12.5% incidence of LS was found, which agrees with the research literature. Nonetheless, the analysis of statistical significance in these results has to be evaluated with a higher number of cases in order to justify the real incidence of LS in this medium. Further to this point, in clinical practice LS has been diagnosed little due especially to the lack of specific characteristics of the disease in most cases, apart from phimosis, that is. We should like to add the fact that its incidence is uncertain, due to the lack of routine in the anatomopathologic analysis of the excised prepuces in the phimosis cases.20
Another anatomopathologic alteration that could be verified was lichenoid infiltrate, found in 18% of patients. Ackerman27 includes this alteration as one of the initial histopathologic findings of LS. Therefore, as the lichenoid infiltrate is possibly associated with LS, one cannot exclude the possibility that these cases belong to an evolutive pathological process whose final result is LS. Such a possibility would considerably increase the incidence of phimosis as caused by LS.
With respect to treating phimosis, the literature points to 80% effectiveness with topical corticosteroid use in phimosis.6,29 Likewise, some authors have shown that topical corticosteroids are active on treating LS at the initial and intermediary stages; at more advanced stages of the disease, circumcision is the chosen treatment.10,24 The aforementioned treatment with topical corticosteroids was utilized with 70% of patients analyzed in this paper. LS cases represent 14% of the cases whose treatment failed. Nonetheless, such data are hardly the most precise given that some patients undergo treatment only for a few days.
Failure of clinical treatment with corticotherapy was the main indication for surgery in this casuistry, with 45% of cases. This difference with the literature, which shows 80% effectiveness, cannot be valorized due to the fact that the authors did not follow up this prior treatment. The latter situation thereby makes it impossible to figure out whether the procedure was performed correctly, namely, with the objective of obtaining data in order to comparing them with the literature. The other surgical indications were: recurrent balanoposthitis, with 25% of indications; the clinical condition of (severe) narrowing of the prepuce, with 17.5% incidence; recurrent UTI, 10%; and associated urinary diseases, 2.5%. The urinary disease found in the latter instance was a case of lithiasis of the lower urinary tract.
There were no complications resulting from the treatment performed, independently of the diagnosis of LS, which confirms postectomy as the definitive treatment for this condition.31
1. The histologic characteristics of phimosis are varied. They do not show a single pattern.
2. Among the histopathologic findings, the absence of alterations, lichenoid pattern and specific alterations of LS were featured.
3. In most cases, the histopathologic findings are inexpressive, i.e. without showing any significant alterations.
4. If the lichenoid patterns encountered in some cases were considered to be initial manifestations of LS, this would significantly increase LS incidence as a cause of phimosis.
5. Lichen sclerosis as a cause of phimosis in children showed an incidence of 12.5% in the sample studied.
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R. Prof. Luis Bezerra da Trindade, n. 106 - Apto 101
88015-160 Florianópolis SC
Tel/Fax: (48) 222-9987 / 223-5784
in January, 27th of 2003.
Approved by the Consultive Council and accepted for publication in October, 16th of 2003.
* Work done at "Hospital Infantil Joana de Gusmão e Instituto Diagnóstico de Anatomia Patológica - IDAP, Florianópolis".