Introduction: A peculiar spongy appearance of the perianal skin was observed in a patient who underwent wide excision surgery for inguinal and gluteal hidradenitis suppurativa (HS). Case Presentation: This peculiar appearance was observed in a 62-year-old male patient. It included multiple orifices and cavities forming the spongy aspect of the perianal skin with multiple cysts and giant comedones. The perianal lesions were asymptomatic and the patient had never received any perianal treatment for the comedones or cysts. Histopathology was performed and demonstrated dilated hair follicles with flaky keratin and loose hair shafts in the center. The spongy appearance was most likely the result of spontaneous shedding of the cystic contents. Conclusion: We suspect that the “cystic sponge anus” might be associated with HS, smoking, the male gender, and may yet be another expression of an occlusive follicular disease. Future studies will be needed to clarify the prevalence and comorbidities of the “cystic sponge anus.”

Hidradenitis suppurativa (HS) is a chronic auto-inflammatory skin disease with typical painful lesions, such as inflammatory nodules, abscesses, and draining tunnels [1]. These lesions predominantly occur in the intertriginous body areas and can be accompanied by less typical lesions, such as pustules, ulcers, comedones, and epidermal cysts [2]. In this case however, we noticed a formation of unusual lesions of the perianal skin. For this case report, the CARE checklist has been completed by the authors, attached as supplementary material (for all online suppl. material, see https://doi.org/10.1159/000536085).

A 62-year-old male underwent wide excision surgery for inguinal and gluteal HS. This patient had multiple inflammatory nodules and draining tunnels in the inguinal and gluteal region. The first HS symptoms started at the age of 14 and he received multiple courses of antibiotics, such as clindamycin and metronidazole, and three prior surgical interventions. The patient was an active smoker with a BMI of 25.7 kg/m2 at the time of the surgery. During surgery, we observed multiple orifices and cavities forming a peculiar spongy appearance of the perianal skin with multiple cysts and giant comedones (Fig. 1).

Fig. 1.

This picture of the perianal region shows multiple small orifices and cavities that form the sponge aspect of the skin. Furthermore, it shows multiple perianal cysts and giant comedones.

Fig. 1.

This picture of the perianal region shows multiple small orifices and cavities that form the sponge aspect of the skin. Furthermore, it shows multiple perianal cysts and giant comedones.

Close modal

The perianal lesions were asymptomatic and the patient had never received any perianal treatment for the comedones or cysts. Histopathology was performed and demonstrated dilated hair follicles with flaky keratin and loose hair shafts in the center. Therefore, we concluded that this spongy aspect most likely is the result of spontaneous shedding of the cystic contents.

Reports on multiple perianal cysts are scarce and to our knowledge, this additional spongy phenomenon has not been described before. However, it should be noted that these cases reported no characteristics of HS [3, 4]. We suspect that the “cystic sponge anus” might be associated with HS, smoking, the male gender, and may yet be another expression of an occlusive follicular disease. Furthermore, we see a resemblance with previously reported cases with multiple vellus hair cysts in the perianal region [4]. In our case however, histopathology was only conducted on the cavities, so a comparison with the vellus hair cysts is not possible. Future studies will be needed to clarify the prevalence and comorbidities of the “cystic sponge anus.”

This study was conducted in accordance with the provisions of the Declaration of Helsinki. Written informed consent was obtained from the patient for publication of the details of their medical case and any accompanying images. Consent for the publication of all patient photographs and medical information was provided by the authors at the time of article submission to the journal stating that all patients gave consent for their photographs and medical information to be published in print and online and with the understanding that this information may be publicly available. This retrospective review of patient data did not require ethical approval in accordance with local/national guidelines.

The authors have no conflicts of interest to declare.

No funding was received for this study.

Conceptualization: Hessel van der Zee; data curation and writing – original draft: Pim Aarts; writing – review and editing: all authors.

All data generated or analyzed during this study are included in this article. Further inquiries can be directed to the corresponding author.

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