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A rare case of syringocystadenoma papilliferum: clinical and histopathological insights Andayani, Raden Roro Rini; Pudjiati, Satiti Retno; Harsono, Erliana Tantri; Rizkiani, Dwinanda Almira; Hanggoro Tri Rinonce; Rita Cempaka; Indra Wahono; Siti Isya Wahdini; Dyah Ayu Mira Oktarina
Indonesian Journal of Biomedicine and Clinical Sciences Vol 57 No 3 (2025)
Publisher : Published by Universitas Gadjah Mada

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.22146/inajbcs.v57i3.17146

Syringocystadenoma papilliferum (SCAP) is a rare benign adnexal neoplasm originating from apocrine or eccrine sweat glands, first reported by Stokes in 1917. Clinically, SCAP manifests in various forms such as nodules, plaques, or warts, predominantly affecting the scalp in adolescents or children. Histopathologically, it is characterized by papillary and ductal invaginations lined with a two-layered epithelium. This condition can be present at birth or emerge during puberty. We present a 19-year-old female with a congenital, pinkish-brown, wart-like lesion on her scalp that progressively enlarged and spread to the face. Physical examination revealed a verrucous plaque with central erosion, extending from the temporal scalp to the lateral face. Dermoscopy showed papillary structures with defined lesion borders, while histopathological findings confirmed SCAP. The patient underwent surgical excision, and no new lesions were detected during a 3 mo follow-up. Syringocystadenoma papilliferum is often confused with other neoplasms such as sebaceous nevus or verruca vulgaris, but its definitive diagnosis relies on histopathology. The tumor’s characteristic features include cystic invaginations into the dermis with a two-layered epithelium, differentiating it from other conditions. Surgical excision remains the treatment of choice to prevent potential malignant transformation and improve cosmetic outcomes. Syringocystadenoma papilliferum, though rare, can be effectively managed through timely diagnosis and surgical excision. In this case, the patient’s postoperative outcome was favorable, with no recurrence of the lesion. Histopathological evaluation is crucial for accurate diagnosis and guiding treatment strategies.

Histopathology as a key to identify Sezary syndrome in patient with clinical erythroderma Diovani, Sonia; Irianiwati; Febriana, Sri Awalia; Waskito, Fajar; Rizkiani, Dwinanda Almira; Andayani, Raden Roro Rini; Rifko, Ani
Indonesian Journal of Biomedicine and Clinical Sciences Vol 57 No 3 (2025)
Publisher : Published by Universitas Gadjah Mada

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.22146/inajbcs.v57i3.17293

Sezary syndrome (SS) is a rare and aggressive variant of cutaneous T-cell lymphoma (CTCL), characterized by clonal proliferation of malignant T lymphocytes with skin erythroderma. The incidence of CTCL in the United States from 2000 to 2010 reached 10 cases per million people per year, accounts for only 3% of all cases of cutaneous lymphoma. In Indonesia, lymphoma ranks sixth in terms of malignancies, alongside Hodgkin lymphoma and leukemia. We reported a case of 56 y.o. woman presented with blistering lesions that turned into wounds all over her body. Skin biopsy examination revealed characteristic histopathological features, including basket weave-type orthokeratosis, focal parakeratosis, focal acanthosis, and epidermotropism of atypical lymphoid cells with cerebriform nuclei (Sezary cells). Immunohistochemical examination using CD3, CD4, and CD8 showed positive staining in the membrane and cytoplasm of tumor cells within the dermis and epidermis. Sezary syndrome is characterized by clonal expansion of T-helper memory cells in the skin. Histopathology and immunohistochemistry could not differentiate SS from mycosis fungoides. However, histopathological examination could distinguish SS from other differential diagnoses. The diagnosis of SS can be established based on the triad of 1) generalized exfoliative dermatitis involving more than 80% of the body surface area, 2) lymphadenopathy, and 3) the presence of 5% or more malignant T-cells with cerebriform nuclei (Sezary or Lutzner cells) in peripheral blood lymphocytes. In conclusion, we present a case of SS, diagnosed based on the patient's history, physical examination, peripheral blood examination, histopathology, and immunohistochemistry.

Diagnostic challenges and clinical insight of medial thigh hemangiolymphangioma in adult: A rare case report Rizkiani, Dwinanda Almira; Trisnowati, Niken; Khalidah, Miya; Anggatama, Marcella; Dyah Ayu Mira Oktarina
Indonesian Journal of Biomedicine and Clinical Sciences Vol 57 No 4 (2025)
Publisher : Published by Universitas Gadjah Mada

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.22146/inajbcs.v57i4.17129

Vascular anomalies are classified into vascular tumors and vascular malformations. Hemangiolymphangioma (HLA) is a rare vascular malformation that contains both blood and lymphatic components, most commonly diagnosed in infancy or early childhood. Adult cases, especially in atypical locations such as the medial thigh, are exceedingly rare and pose diagnostic challenges. A 27 yo female presented with a slowly enlarging, painless lump on the left medial thigh. Initially misdiagnosed as condyloma acuminata, the lesion was subsequently identified as a vascular malformation through dermoscopic visualization of characteristic vascular lacunae. Histopathological and immunohistochemical analyses confirmed the diagnosis of HLA. Surgical excision was performed with clear margins, and no recurrence was observed after 3 mo. Hemangiolymphangiomas are benign but may exhibit local infiltration and recurrence, especially after incomplete resection. Diagnosis requires a multimodal approach including clinical assessment, dermoscopy, histopathology, and immunohistochemistry. Differential diagnosis includes hemangioma, lymphangioma, and malignancies such as lymphangiosarcoma. Complete surgical excision remains the treatment of choice, with other modalities like electrocautery or cryotherapy considered in selected cases. Long-term follow-up is crucial due to the risk of recurrence. In conclusion, this rare adult case of medial thigh HLA highlights the importance of considering vascular malformations in atypical anatomical sites. Early recognition and comprehensive diagnostic evaluation facilitate appropriate management and improve patient outcomes

Histopathology as a key to identify Sezary syndrome in patient with clinical erythroderma Diovani, Sonia; Irianiwati; Febriana, Sri Awalia; Waskito, Fajar; Rizkiani, Dwinanda Almira; Andayani, Raden Roro Rini; Rifko, Ani
Indonesian Journal of Biomedicine and Clinical Sciences Vol 57 No 3 (2025)
Publisher : Published by Universitas Gadjah Mada

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.22146/inajbcs.v57i3.17293

Sezary syndrome (SS) is a rare and aggressive variant of cutaneous T-cell lymphoma (CTCL), characterized by clonal proliferation of malignant T lymphocytes with skin erythroderma. The incidence of CTCL in the United States from 2000 to 2010 reached 10 cases per million people per year, accounts for only 3% of all cases of cutaneous lymphoma. In Indonesia, lymphoma ranks sixth in terms of malignancies, alongside Hodgkin lymphoma and leukemia. We reported a case of 56 y.o. woman presented with blistering lesions that turned into wounds all over her body. Skin biopsy examination revealed characteristic histopathological features, including basket weave-type orthokeratosis, focal parakeratosis, focal acanthosis, and epidermotropism of atypical lymphoid cells with cerebriform nuclei (Sezary cells). Immunohistochemical examination using CD3, CD4, and CD8 showed positive staining in the membrane and cytoplasm of tumor cells within the dermis and epidermis. Sezary syndrome is characterized by clonal expansion of T-helper memory cells in the skin. Histopathology and immunohistochemistry could not differentiate SS from mycosis fungoides. However, histopathological examination could distinguish SS from other differential diagnoses. The diagnosis of SS can be established based on the triad of 1) generalized exfoliative dermatitis involving more than 80% of the body surface area, 2) lymphadenopathy, and 3) the presence of 5% or more malignant T-cells with cerebriform nuclei (Sezary or Lutzner cells) in peripheral blood lymphocytes. In conclusion, we present a case of SS, diagnosed based on the patient's history, physical examination, peripheral blood examination, histopathology, and immunohistochemistry.

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