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  • Research article
  • Open Access

Penile Lymphangioma: review of the literature with a case presentation

  • 1,
  • 1,
  • 2 and
  • 3Email author
Contributed equally
Basic and Clinical Andrology201929:1

https://doi.org/10.1186/s12610-018-0081-3

  • Received: 22 August 2018
  • Accepted: 5 December 2018
  • Published:

Abstract

Background

Penile lymphangiomas are rare manifestations of lymphangiomas or lymphatic malformations which are more commonly found in the head or neck region of the body. Lymphangiomas are further categorized as lymphangioma circumscriptum, cavernous lymphangioma, cystic hygroma, or acquired lymphangiomas (also known as lymphangiectasia), based on their depth and etiology.

Results

A literature review revealed only 30 cases of penile lymphangioma between 1947 and March 30, 2018. Several causes were attributed to the acquired penile lymphangiomas, including trauma, phimosis, and infection. While penile lymphangiomas can be initially mistaken for an infection, a thorough history and physical examination is sufficient to clinically diagnose a lymphangioma of the penis. Historically, surgical excision has been the gold standard of treatment for this condition. When asymptomatic, patients may opt for conservative management with avoidance of mechanical trauma alone. Other physicians have revealed novel treatment plans to rid patients of their penile lymphangioma such as a staged laser procedure.

Conclusion

In this article, we elucidate the causes, symptoms, treatments, and outcomes associated with penile lymphangiomas found in the literature while also presenting the case of a 30-year-old African-American man diagnosed with acquired penile lymphangioma.

Résumé

Contexte

Les lymphangiomes du pénis sont des manifestations rares des lymphangiomes ou des malformations lymphatiques qui sont plus fréquemment observés dans les régions de la tête ou du cou au niveau du corps. Les lymphangiomes sont aussi classés en lymphangiome circumscriptum, lymphangiome caverneux, hygroma kystique, ou en lymphangiomes acquis (aussi connus sous l’appellation lymphangiectasie), selon leur profondeur et leur étiologie.

Résultats

Une revue de la littérature ne révèle que 30 cas de lymphangiomes du pénis rapportés entre 1947 et Mars 2018. Plusieurs causes ont été attribuées aux lymphangiomes acquis du pénis, parmi lesquelles les traumatismes, le phimosis et les infections. Bien que les lymphangiomes péniens puissent être initialement pris pour une infection, une recherche des antécédents et un examen clinique minutieux sont suffisants pour diagnostiquer cliniquement un lymphangiome du pénis. L’excision chirurgicale a toujours constitué le gold standard du traitement de cette maladie. Lorsqu’elle est asymptomatique, les patients peuvent choisir un traitement conservateur en évitant les seuls traumatismes mécaniques. D’autres médecins ont proposé de nouveaux traitements ayant pour objectif de débarrasser les patients de leurs lymphangiomes péniens, comme la procédure au laser par étapes.

Conclusion

A partir des données de la littérature, nous avons clarifié dans cet article les causes, symptômes, traitements et résultats associés aux lymphangiomes du pénis ; nous avons aussi présenté le cas d’un homme afro-américain âgé de 30 ans diagnostiqué comme porteur d’un lymphangiome pénien acquis.

Mots-clés

Caverneux, Circumscriptum, Kystique, Hygroma, Lymphangiome, Lymphangiectasie, Pénien, Pénis.

Keywords

  • Cavernous
  • Circumscriptum
  • Cystic
  • Hygroma
  • Lymphangioma
  • Lymphangiectasis
  • Lymphangiectasia
  • Penile
  • Penis

Background

Redenbacher et al. first described lymphangiomas, or “lymphatic malformations,” in 1828 [1]. Since then, lymphangiomas have been classified further in the literature as lymphangioma circumscriptum, cavernous lymphangiomas, cystic hygromas, and acquired lymphangiomas (also known as lymphangiectasias). Lymphangiomas are most commonly found in the head and neck, and two-thirds of all lymphangiomas are found by two years of age. The literature provides detailed pathological, radiographical, and clinical findings associated with lymphangiomas of the head and neck. On the contrary, fewer than 50 cases of lymphangiomas of the penis have been reported since the first description by Ferris et al. in 1944. Presumably, lymphangiomas of the penis are under-reported because the penile lesions, unlike the pathological counterparts in the head/neck, often go unnoticed by not only the physician but also the patient [2]. Furthermore, lymphangiomas of the penis are often misdiagnosed and mistreated as genital warts, molluscum contagiosum or gonorrhea [3]. The objective of this study is to define the different types of penile lymphangiomas by etiology, clinical findings, and treatments. Also, we report a case of acquired lymphangioma of the penis in a 30-year-old male treated successfully by surgical excision.

Methods

We conducted a literature review identifying all publications using the keywords: “penile” OR “penis” AND “lymphangioma” OR “lymphangiectasis” OR “lymphangiectasia.” Following our institutional protocol, the literature review identified relevant studies via a computer-aided search of American (MEDLINE from 1946 – March 31, 2018) and European articles (Embase 1947 – March 31, 2018). Publications found in languages other than English were excluded. Publications reporting lymphangiomas of the scrotum, perineum, and/or surrounding area not including the penis were excluded. The publications reporting benign transient lymphangiectasis of the penis (BTLP) were also excluded as BTLP, commonly known as sclerosing lymphangitis, is a transient condition that often results after sexual activity and holds minimal medical or surgical relevance. Classifications of penile lymphangioma found in Table 1 corresponding to the case classification from their original publication include “Acquired” (lymphangioma), “Cavernous” (lymphangioma), (lymphangioma) “Circumscriptum,” and (lymphangioma) “Circumscriptum Cysticum” (for cystic hygroma). The classification “Acquired [inferred]” was reserved for cases of lymphangiectasis/lymphangiectasia as well as reported cases of lymphangioma circumscriptum resulting from non-congenital causes. Although controversy exists in the difference between lymphangioma and lymphangiectasis/lymphangiectasia, all cases of lymphangiectasis/lymphangiectasia are reported as “Acquired [inferred]” in Table 1 for simplicity.
Table 1

A summary of penile lymphangioma cases reported in the literature

Author

Diagnosis

Age of Onset

Causes/Comorbidities

Location

Treatment

Outcome

Present case

Acquired

30 years old with 21-year history

Skin of penis caught in zipper when patient was 9 years old

Asymptomatic bumps circumferentially around distal shaft of penis

Surgical resection

No pain, erythema or discharge found at 1 month follow-up

Gupta S et al. [11]

Acquired

20 years old

Recurrent swelling with multiple, minute, papulo-vesicular lesions in right foot and leg from age of 3–4 months

Asymptomatic papular lesions on penis and scrotum present for 2–3 months

Simple electrofulguration of visible papulo-vesicles on penis

Acquired

35 years old with 20-year history

Papular lesions and gradual swelling of scrotum and penis (shaft, frenulum and around external urethral meatus)

No intervention

S Adikari et al. [10]

Acquired

47 years old with 25-year history

Misdiagnosed as genital warts, treated for gonorrhea 5 years after

Smooth to palpation, wart-like lesion on dorsal aspect of penis, otherwise asymptomatic

Surgical excision

Successful with no sign of reccurrence

D. M. Piernick 2nd et al. [8]

Acquired

48 years old with 5-year history

Hidradenitis suppurativa of buttocks, gluteal cleft and perineal area

Asymptomatic, multiple semitranslucent skin colored papules coalescing into plaques on penile shaft, scrotum and perineum

None

Errichetti et al. [4]

Acquired

61 years old with 1-year history

Severe phimosis

Constricting phimotic ring and considerable edema of glans and distal foreskin with several translucent preputial papulovesicles (some slightly hyperkeratotic) localized close to balanopreputial sulcus

None - patient was waiting for surgery to correct phimosis – no follow-up information was provided

Zhang et al. [6]

Acquired

8 years old with 2-week history

Surgery to correct phimosis

Asymptomatic, multiple small vesicular lesions on glans

“watch and wait” policy

Lesions resolved in 3 weeks

Dehner LP. et al. [21]

Acquired

39 years old with 6-week history

Shaft, dorsum of penis

Surgical excision

Successful with no sign of recurrence

Ferris et al. [2]

Acquired

35 years old with 30-year history

Measles? Circumcision? Pneumonia? All illnesses exacerbated condition

Lesions on foreskin of penis, scrotum and adjacent areas of the thigh and perineum

Surgical excision with skin graft

Successful with no sign of recurrence

Hagiwara et al. [9]

Acquired

65 years old with 18-year history

Filariasis

Scrotum, extending to foreskin

Surgical excision and skin grafting

Successful with no sign of recurrence - transient penile edema present for few weeks

Sadikoglu et al. [5]

Acquired (inferred)

15 years old with 3-year history

Blunt trauma caused skin thickening

Penile and scrotal skin

Surgical excision and skin grafting

Successful with no sign of recurrence

Kokcam et al. [19]

Acquired (inferred)

19 years old with 3-year history

Multiple translucent and hemorrhagic vesicles on shaft and glans of penis. Surface was smooth, some umbilicated

Pt refused surgical intervention, advised to avoid mechanical trauma, apply silver sulfadiazine cream to ruptured lesions

No new lesions, overall number of lesions declined markedly with no other complications

Latifoglu et al. [22]

Acquired (inferred)

10 years old with 6-year history

Penoscrotal lymphedema with erythematous plaque (irregular, well-defined border) on penile shaft and gelatinous-appearing, coalescent, verrucous vesicles and papules on scrotum

Surgical resection

Successful with no sign of recurrence

Maloudijan et al. [26]

Acquired (inferred)

50 years old with 10-year history

Asymptomatic, 2 mm large vesicular lesions in sulcus coronarius from adjacent foreskin and glans

Patient abstained

Cestaro et al. [23]

Acquired (inferred)

24 years old

HPV - genotype 6 comorbidity

Lesions on inguinal area, scrotum, penis, glans with associated edema of penis and lips

Surgery

Successful with no recurrence

Shi G. et al. [7]

Acquired [inferred]

23 years old with 40-day history

Circumcision following phimosis 5 years ago

Asymptomatic translucent, yellowish, elevated, thick walled cystic lesions on right side of glans

2940 nm nonablative fractional Er:YAG laser at 2–3 week intervals with power density of 3 J/cm2 at 20 ms and a 5 mm spot size

Lesions disappeard obviously after 4 sessions, no recurrence, dyspigmentation and paresthesia

Shah A. et al. [24]

Acquired [inferred]

11 years old with few month history

Asymptomatic, soft mass on dorsal aspect of penis with extension towards right hemiscrotum

Local surgical resection

Recurrence 11 months following surgery

Bardazzi et al. [25]

Acquired [inferred]

45 years old

Sulcus of prepuce

Diathermy

Successful with no sign of recurrence

Llanes et al. [16]

Cavernous

20 years old

Soft lesion in dorsal area of prepuce

Circumcision

Successful with no sign of recurrence

Hayashi et al. [17]

Cavernous

32 years old

Tumor on coronary sulcus of glans and submucosa

Cavernous

35 years old

Tumor on coronary sulcus of glans and submucosa

Geuekdjian et al. [27]

Circumscriptum

3 years old

Congenital [inferred]

Asymptomatic, edematous swelling of penis particularly in skin spreading upwards to left groin

En bloc resection

Successful with no sign of recurrence

Demir et al. [18]

Circumscriptum

21 years old with history since childhood

Congenital [inferred]

Recurrent infections, drainage of vascular lesions, penoscrotal deformity and inability to have sexual intercourse

Surgical excision

Successful, no sign of remission

Ferro et al. [3]

Circumscriptum

16 years old

Congenital [inferred]

Tense vesicles filled with clear fluid on coronal region

3 surgeries - remission every time. Denuded penis buried in tunnel guided through scrotum, 6 months after - shaft lift and recreated with scrotal skin

No negative consequences, local hairiness treated cosmetically

Osborne et al. [14]

Circumscriptum

45 years old

Lichen planus - treated with cryotherapy

Cluster of translucent vesicles on shaft of penis and coronal sulcus. Balanomegaly.

Treatment declined

Tsur et al. [28]

Circumscriptum

8 month old

Congenital [inferred]

asymptomatic elevated lesions on glans penis around meatus and dorsal aspect of penis

Surgical excision

Successful with no sign of recurrence

Drago et al. [12]

Circumscriptum

27 years old

Ulceritive colitis

 

Handa et al. [29]

Circumscriptum

10 years old with 9 year history

Congenital [inferred]

Penis, scrotum, groins bilaterally

Swanson et al. [13]

Circumscriptum

16 years old

Recurrent cellulitis of the penis and scrotum

Subcutaneous tissue of penis proximal to glans and skin of left proximal scrotum

Greiner et al. [15]

Circumscriptum cysticum

13 years old

Congenital malformation

Edematous thickening of penile and scrotal skin

A summary of penile lymphangioma cases reported in the literature

Case presentation

A 30-year-old African-American male presented to his primary care physician with a chief complaint of a several-year history of unhealing wounds on the right side of his penile shaft after his penis was caught in his zipper several years ago. Patient became concerned after noting white penile discharge 2 weeks prior. He denies any anal or oral lesions as well as exposure to any sexually transmitted diseases (STDs). Following outpatient specialty referral, the dermatologist reported excess skin tissue with underlying edema circumferentially on the distal penile shaft with overlying multiple firm skin-colored papules, some with exophytic crusting [Fig. 1]. Chlamydia trachomatis, human immunodeficiency virus (HIV), Neisseria gonorrhea and syphilis were negative. Subsequent biopsy found dilated vascular channels consistent with benign acquired lymphangioma of the penis (Fig. 2), and the patient was referred to urology for evaluation and management. With the urologist, the patient elected for surgical intervention due to cosmetic concerns despite the asymptomatic nature of the lymphangioma.
Fig. 1
Fig. 1

Clinical examination of patient’s penile shaft and glans: Multiple firm skin-colored papules, some with exophytic crusting and underlying edema, present on the right side of the patient’s penile shaft, immediately proximal to the glans

Fig. 2
Fig. 2

Histopathological image following biopsy of patient’s penile lesions: Histopathological staining from biopsy by punch technique of distal dorsal penile shaft shows dilated vascular channels consistent with benign lymphangioma of the penis

The patient underwent circumcision for redundant prepuce, excision of the skin lesion and penile foreskin reconstruction. A circumferential incision was made on the mucosa 0.5 cm proximal to the glans, distal to the lymphangioma. The foreskin was then retracted and another circumferential incision was made around the mucosal skin. The foreskin was then dissected using Bovie cautery and blunt dissection while the foreskin with lymphangioma tissue was excised. Intraoperative and postoperative courses were unremarkable. At 1-month follow-up, the patient reported no pain, erythema or discharge from the wound.

Results

The current literature review identified 27 cases in 25 publications. In the largest group, the 18 acquired lymphangiomas of the penis identified in the literature review encompassed a wide variety of cases. One was attributed to phimosis and 2 to trauma [4, 5]. Two iatrogenic cases resulted from correction of phimosis [6, 7]. One infectious case was attributed to filariasis (1 case), and 1 inflammatory case was attributed to hidradenitis suppurativa (1 case) [8, 9]. Two idiopathic cases were previously mistaken for genital warts and gonorrhea [10, 11].

Lymphangioma circumscriptum of the penis in this literature review was reported in eight, five of which were ruled to be congenital. The other three were notable for comorbidities of ulcerative colitis, recurrent cellulitis, and Lichen planus [1214]. The only case of lymphangioma circumscriptum cysticum or cystic hygroma was found to have a congenital origin [15]. Lastly, cases of cavernous lymphangioma were attributed to a tumor (2 cases) and one with an unknown cause [16, 17].

Discussion

Lymphangiomas are generally described as uncommon, hamartomatous malformations of the lymphatic system. Based on depth and size of the lymph vessels, classifications include lymphangioma circumscriptum (most superficial), cavernous lymphangioma and cystic hygroma (most deep). Further subdivisions derive from perceived cause. Congenital lymphangiomas are thought to result from fetal lymph vessels that failed to involute and/or failed to join with the central lymphatic system. On the contrary, acquired lymphangiomas may result from trauma, certain infections (cellulitis, neoplastic disease, tuberculosis, filariasis), radiotherapy, pregnancy, scleroderma, severe phimosis or STDs [10].

Although the causes of these various types of lymphangioma may differ, the clinical presentation is often similar. The majority of the penile lymphangiomas present as asymptomatic, fluid-filled, translucent lesions or vesicles most commonly on the shaft or coronal sulcus of the penis. Symptomatic lesions, on the other hand, typically focus on sexual dysfunction. One case of acquired lymphangioma presented with severe phimosis and inability to produce an erection [4]. Also, one case of lymphangioma circumscriptum caused recurrent infection, intermittent drainage and sexual inactivity [18]. Given the similarities in clinical presentation, a proper diagnosis becomes contingent on a thorough history and physical examination in the case of lymphangioma of the penis. To that end, clinicians seeking to properly diagnose and treat lymphangiomas of the penis must effectively rule out certain infectious diseases such as molluscum contagiosum and gonorrhea [19]. This is generally possible following a thorough history and physical examination alone. However, in some rare cases, a biopsy with accompanying pathology report or an infectious disease workup may be necessary. Dermatology consultation may be selected for unclear cases. Interestingly, Errichetti et al. recently elucidated the potential role of dermoscopy in the diagnosis of penile lymphangioma by describing the presence of “yellowish-reddish, well-demarcated, round or oval lacunae surrounded by whitish areas or lines,” which may be common characteristics of lymphangiomas [4, 20]. This observation warrants further investigation into the utility of dermoscopy as a quick, non-invasive method of definitively diagnosing lymphangiomas.

Many authors may argue that lymphangiomas of the penis do not always require treatment, given the mostly asymptomatic nature of these lesions. However, patients may request intervention for cosmetic reasons. Of the 18 acquired lymphangiomas reported in Table 1 (including the present case), 9 were treated by surgical excision [2, 5, 9, 10, 2123] – only one of which recurred 11 months following surgery [24]. And, one case reported an electrofulguration (or electrocautery) of the visible papulo-vesicles on the penis [11]. Shi et al. found that the acquired lymphangioma of the penis was amenable to 2940-nm Erbium-doped Yttrium Aluminum Garnet laser once every 2–3 weeks, wherein the lesions disappeared after the fourth session with no evidence of recurrence, dyspigmentation or paresthesia [7]. Bardazzi et al. utilized high-frequency electric currents, called diathermy, to successfully obliterate the acquired lymphangioma without evidence of recurrence [25]. On the other hand, Zhang et al. advocated for a “watch and wait” policy that interestingly allowed the lesions to self-heal within three weeks time [6]. Lymphangiomas that develop following circumcision may spontaneously resolve. Abstinence from treatment was observed in two patients, one of whom opted for protecting the lesions from mechanical trauma and applying a silver sulfadiazine cream to any ruptured lesions [19, 26]. This management decreased the number of existing lesions and prevented new lesions. Finally, one patient was waiting for surgery to correct phimosis; no follow-up information was provided [4]. Neither treatment nor follow-up was documented in the remaining two cases [8, 11].

Of the 3 cavernous lymphangiomas reported in Table 1, one underwent circumcision with no sign of recurrence following the treatment [16]. The other two cases failed to specify treatment [17]. Of the 8 cases of lymphangioma circumscriptum of the penis, 3 underwent surgical resection successfully with no signs of recurrence [18, 27, 28]. Another patient with lymphangioma circumscriptum (Table 1) underwent 3 surgeries with recurrence every time. For the fourth operation the physicians adopted a more radical surgical approach, in which the penile shaft was denuded and buried in the scrotum [3]. Six months later, the penile shaft was raised, and the skin was reconstructed using a scrotal graft. The outcome of this procedure was favorable with the only noteworthy complication of growth of transposed hair, treated cosmetically. Of the remaining 4 lymphangioma circumscriptum cases shown in Table 1, treatment was denied for 1, and the remaining 3 had no information on treatment or outcome [1214, 29].

Conclusions

In summary, penile lymphangioma can be divided into four categories: 1) acquired, 2) lymphangioma circumscriptum, 3) cavernous lymphangioma, and 4) cystic hygroma. Commonly mistaken for infectious lesions, lymphangiomas underscore the importance of an appropriate history and physical to properly identify and treat the lymphatic malformation.

Notes

Abbreviations

BTLP: 

Benign transient lymphangiectasis of the penis

HIV: 

Human immunodeficiency virus

STDs: 

Sexually transmitted diseases

Declarations

Acknowledgments

Not applicable.

Funding

There are no funding sources to report.

Availability of data and materials

The datasets used and/or analyzed for this study can be procured from the corresponding author upon request.

Author’s contributions

MM assisted in manuscript composition and final review. SA assisted in manuscript composition and data collection. *MM and SA contributed equally to this submission. Hayan Jaratli provided the images and histology description. AD edited the manuscript and confirmed the accuracy of manuscript content. All authors read and approved the final manuscript

Ethics approval and consent to participate

This study was approved by the Henry Ford Hospital Institutional Review Board (IRB #11419).

Consent for publication

Upon outpatient follow-up, the patient consented to publishing his information and images (although they have been anonymized). The appropriate forms have been uploaded.

Competing interests

The authors declare that they have no competing interests.

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Authors’ Affiliations

(1)
Department of Neurosurgery, Henry Ford Hospital, 2799 West Grand Blvd, Detroit, MI 48202, USA
(2)
Department of Pathology, Henry Ford Hospital, 2799 West Grand Blvd, Detroit, MI 48202, USA
(3)
Department of Vattikuti Urology Institute, Henry Ford Hospital, 2799 West Grand Blvd, Detroit, MI 48202, USA

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Copyright

© The Author(s). 2019

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