Breast implant-associated anaplastic large cell lymphoma: two distinct clinicopathological variants with different outcomes.

BACKGROUND: ALK-negative anaplastic large cell lymphoma associated with breast implant (i-ALCL) has been recently recognized as a distinct entity. Among 43 830 lymphomas registered in the French Lymphopath network since 2010, 300 breast lymphomas comprising 25 peripheral T-cell lymphomas (PTCL) were reviewed. Among PTCL, ALK-negative ALCL was the most frequent and all of them were associated with breast implants. PATIENTS AND METHODS: Since 2010, all i-ALCL cases were collected from different institutions through Lymphopath. Immuno-morphologic features, molecular data and clinical outcome of 19 i-ALCLs have been retrospectively analyzed. RESULTS: The median age of the patients was 61 years and the median length between breast implant and i-ALCL was 9 years. Most implants were silicone-filled and textured. Implant removal was performed in 17 out of 19 patients with additional treatment based on mostly CHOP or CHOP-like chemotherapy regimens (n = 10/19) or irradiation (n = 1/19). CHOP alone or ABVD following radiation without implant removal have been given in two patients. The two clinical presentations, i.e. effusion and less frequently tumor mass correlated with distinct histopathologic features: in situ i-ALCL (anaplastic cell proliferation confined to the fibrous capsule) and infiltrative i-ALCL (pleomorphic cells massively infiltrating adjacent tissue with eosinophils and sometimes Reed-Sternberg-like cells mimicking Hodgkin lymphoma). Malignant cells were CD30-positive, showed a variable staining for EMA and were ALK negative. Most cases had a cytotoxic T-cell immunophenotype with variable T-cell antigen loss and pSTAT3 nuclear expression. T-cell receptor genes were clonally rearranged in 13 out of 13 tested cases. After 18 months of median follow-up, the 2-year overall survival for in situ and infiltrative i-ALCL was 100% and 52.5%, respectively. CONCLUSIONS: In situ i-ALCLs have an indolent clinical course and generally remain free of disease after implant removal. However, infiltrative i-ALCLs could have a more aggressive clinical course that might require additional therapy to implant removal.

[Telangiectasia during amlodipine therapy]. / Télangiectasies au cours d'un traitement par amlodipine.

BACKGROUND: Calcium inhibitors are recommended as first-line treatment in hypertension. We report the development of telangiectasia on the trunk and upper limbs in a female patient on amlodipine (Amlor(®)) that subsided on treatment discontinuation. PATIENTS AND METHODS: A 63-year-old woman consulted for numerous asymptomatic stellate telangiectasias on her upper trunk and shoulders, with sparing of the face. No Darier's sign was seen and clinical examination was otherwise normal. The patient had been treated with amlodipine (Amlor(®)) and a combination of bisoprolol and hydrochlorothiazide (Lodoz(®)) for 5years for essential hypertension. Laboratory tests, which included serum tryptase assay, were normal. Histological analysis showed a normal epidermis with dilated superficial dermal capillaries and no inflammatory infiltrate or dermal elastosis. Amlodipine was discontinued and replaced with ramipril (an ACE inhibitor). The other treatments were changed over and the telangiectasias regressed spontaneously within several months, with no relapse being seen at one year of follow-up. COMMENTS: Reports of telangiectasia associated with calcium inhibitors most commonly involve the dihydropyridine family, of which amlodipine is a member. Inhibition of muscular contraction induced by this treatment results in vasodilatation, accounting for the associated antihypertensive properties, and this could play a role in telangiectasia formation. The literature contains reports of cases of photo-distributed telangiectasia, suggesting that as well as vasodilatation, calcium inhibitors may in certain cases cause abnormal sensitivity of blood vessels to ultraviolet radiation. The absence of facial lesions rules out the hypothesis of photo-induced lesions in our patient.

[Granulomas induced by injections of leuprorelin acetate (Enantone®)]. / Granulomes induits par des injections de leuproréline acétate (Enantone ®).

BACKGROUND: Gonadorelin analogues (LHRH) are used for the endocrine treatment of prostatic cancer, central early puberty and various gynaecological conditions. Cutaneous adverse events seldom occur. We report a case of injection-site granulomas induced by leuprorelin acetate (Enantone®). CASE REPORT: A 76-year-old man presented with several subcutaneous nodules on his left arm. The nodules were hard but painless. He had received subcutaneous injections of Enantone® for prostatic cancer. Histological examination of a skin biopsy specimen demonstrated granulomatous inflammation with a necrotic centre; screening for an infectious aetiology was negative. Serial sections showed giant cells containing translucent round microspheres in the subcutaneous tissue. Limitation to the leuprorelin acetate injection sites in the arm and detection at histological analysis of microspheres probably bound to an injected product militated in favour of granulomas caused by injections of Enantone®. DISCUSSION: Injection-site granulomas caused by Enantone® are rare. Their formation may depend on the mode of administration: the more superficial the injection, the higher the risk of developing granulomas. The formation of these lesions is probably a foreign body reaction to the excipient.

[Pulmonary epithelioid haemangioendothelioma: two different clinical courses]. / Hémangioendothéliome épithélioïde pulmonaire: à propos de deux observations.

The first case report concerns a 59-year-old man presenting a chronic cough and the second a 23-year-old woman farmer presenting with worsening dyspnoea associated with cough, expectoration and haemoptysis. In the second case, the woman died 7 months after the onset of the respiratory symptoms. In both cases, chest radiography and thoracic CT scans showed multiple, bilateral pulmonary nodules, bronchial endoscopy was normal and surgical biopsy revealed epithelioid haemangioendothelioma. It is a rare primary pulmonary tumour which is usually found in soft tissue, bone or liver. Epithelioid haemangioendothelioma is a vascular tumour usually affecting women less than 40 years of age. The histological appearance is characteristic and may be confirmed by immunohistochemistry. Chemotherapy and radiotherapy are generally ineffective and surgery is sometimes impossible because of the multifocal lesions. Epithelioid haemangioendothelioma is considered a sarcoma without precise histological prognostic criteria. In its pulmonary location it is responsible for death due to respiratory failure in 50% of cases.

[Severe cutaneous toxicity after Pemetrexed as second line treatment for a refractory non small cell lung cancer]. / Toxicité cutanée grave après pemetrexed en traitement de deuxième ligne pour un cancer bronchique.

INTRODUCTION: Pemetrexed is a chemotherapeutic drug with good tolerance, used as first line treatment for malignant pleural mesothelioma in association with cisplatin, and alone as second line treatment in resistant or relapsing non-small cell lung cancer (NSCLC). However, cutaneous toxicity has been described, principally as a rash. Cutaneous toxicity of all grades has been observed in up to 14%, and grade 3 or 4 toxicity in 0.8-1.3% of cases. CASE REPORT: We report the case of an 85 year old man treated for NSCLC presenting 15 days after administration of the second cycle of pemetrexed with cutaneous lesions including erythema, bullae, and desquamation, associated with deterioration in his general condition; a skin reaction corresponding to Lyell's syndrome. Treatment with steroids and gammaglobulins led to local resolution and improvement in his general condition. CONCLUSION: Cutaneous toxicity from pemetrexed should be recognised on account of its potential severity. The appearance of skin lesions is an indication for careful follow-up for evidence of Lyell's syndrome for which intensive management is needed.

[Granulomatous hypodermitis revealing a cutaneous mucormycosis in an immunodepressed patient]. / Hypodermite granulomateuse révélatrice d'une mucormycose chez une malade immunodéprimée.

INTRODUCTION: Mucormycosis is an exceptional opportunist fungal infection, despite the ubiquitous nature of its pathogenic agents. It is sometimes revealed by primary cutaneous involvement and its prognosis is bad in the case of visceral dissemination. Our observation illustrates the need for early diagnosis and treatment of this infection. OBSERVATION: An immunodepressed, 45 year-old woman, had developed necrotic hypodermitis lesions on the lower limbs. The skin biopsy led to the diagnosis of mucormycosis. Despite treatment with liposomal amphotericin, the fungal infection worsened, spread to the organs (lungs and brain) and the patient died. DISCUSSION: This case report underlines the potential severity of mucormycosis, which is presently emerging in the onco-hematological field. The infection is sometimes revealed by inaugural ulcerated and/or necrotic cutaneous lesions. Its diagnosis must be evoked early so that salvage medical-surgical treatment can be initiated.