Metastatic Squamous Cell Carcinoma of the Pleura: A Rare Complication of Hidradenitis Suppurativa

Abstract

Patient: Male, 46Final Diagnosis: Metastatic squamous cell carcinomaSymptoms: Short of breathMedication: —Clinical Procedure: Pleural biopsySpecialty: OncologyObjective:Rare diseaseBackground:Squamous cell carcinoma (SCC), also known as Marjolin ulcer, is a rare complication of hidradenitis suppurativa (HS). Metastatic SCC from HS typically involves the axial skeleton or abdominopelvic viscera. Metastatic disease to the lungs is a rare phenomenon with only three reported cases of lung parenchyma. We present a biopsy proven case of metastatic SCC to the pleura from gluteal HS.Case Report:A 46-year-old male with a history of recently diagnosed Marjolin ulcer secondary to gluteal HS was transferred to our intensive care unit for acute hypoxemic respiratory failure secondary to recurrent pleural effusion. On examination, patient was febrile (38.3°C), normotensive (blood pressure 98/65 mm Hg), tachycardic (116 beats/minute) and tachypneic (40 breaths/minute) with oxygen saturation of 93% on room air. He was in moderate distress requiring endotracheal intubation and mechanical ventilation. Chest examination revealed decreased breath sounds bilaterally and skin examination was significant for 18 cm wide sacral lesion. CT thorax showed bilateral pleural effusions, pleural thickening, and scattered nodular densities within both lungs concerning for metastatic disease. Thoracentesis showed lymphocyte predominant exudate with negative cytology for malignant cells. A video-assisted thoracoscopic surgery (VATS) illustrated thickened pleural rind with histopathology and positive p40 stain consistent with invasive well-to-moderately differentiated keratinizing SCC.Conclusions:SCC arising from HS is rare and metastatic disease to the pleura has not been reported previously. Strong clinical suspicion for malignancy is warranted in patients with advanced HS and evolving pulmonary symptoms despite negative cytology.

Neoplastic Implications in Patients Suffering from Hidradenitis Suppurativa under Systemic Treatments

“…Many authors describe fatal cases of SCC arising in severe long-standing HS of the perineal and perianal sites or of the buttocks, often complicated by inguinal lymph node metastases [ 35 ]. Joglekar et al reported a case of a 46-year-old male with a history of MU secondary to gluteal HS who developed bilateral pleural effusions, pleural thickening and scattered nodular densities within both lungs concerning for metastatic disease; the wide local excision of sacro-gluteal SCC occurred too late in this case, as the patient already had metastatic disease to inguinal lymph nodes before surgery [ 50 ]. SCC of the perineal area metastatising to the inguinal lymph nodes and to the liver metastases was also reported [ 26 ].…”

Multiple SCCs arising in the gluteal area from patient with short disease duration of hidradenitis suppurativa (hurley I‐II): Surgical approach with favorable outcome

“…2,3 It develops three times more often in males, probably due to the more frequent gluteal and perineal localization in males and is highly associated with smoking. [1][2][3][4] The average period between HS onset and SCC developing is estimated of 27 ± 9.9 years, 1,4 significantly longer than in the presented case (~2.5 years). In addition, cases in medical literature report SCC developing in advanced HS, whereas in the presented case the condition is assessed as hurley I-II.…”

“…2 On the other hand, the highly malignant nature of this carcinoma determines its poor prognosis. 1,3 The wide surgical excision has an excellent therapeutic result, showing a recurrence rate of only 2.5%, mainly in advanced forms. 5 Complications are relatively mild such as bleeding and forming of hematoma.…”