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Letter to the Editor
Benign metastatic leiomyomatosis: A case report
Leiomiomatosis metastatizante benigna: presentación de un caso
Anna Soler Sendraa,
Corresponding author
asoler@fphag.org

Corresponding author.
, María del Valle Rodriguez Santasb, Andrea Rodríguez Fernándezc
a Servicio de Medicina Interna, Hospital General de Granollers, Granollers, Barcelona, Spain
b Servicio de Anatomia Patológica, Hospital General de Granollers, Granollers, Barcelona, Spain
c Servicio de Traumatologia, Hospital General de Granollers, Granollers, Barcelona, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Benign metastatic leiomyomatosis is a rare entity of which approximately 100 cases have been described in the medical literature&#46; It usually affects women with a history of hysterectomy&#46; It is characterised by the development of benign tumours corresponding to leiomyomas that spread to other organs&#44; especially the lungs&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of an incidental diagnosis due to SARS-CoV-2 pneumonia&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">A 54-year-old female patient with a history of hysterectomy 20 years earlier due to uterine leiomyoma&#46; She visited the emergency department for acute cough and fever and was diagnosed with SARS-CoV-2 pneumonia&#46; A chest X-ray showed multiple bilateral pulmonary nodules compatible with metastases&#46; For this reason&#44; a chest CT scan was performed&#44; which showed the presence of multiple nodular lesions of different sizes and densities suggestive of a neoplastic process&#46; The work-up was completed with an abdominal CT scan&#44; which detected the presence of solid nodules involving the psoas muscles and a peritoneal nodule in the hypogastrium&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Regarding the SARS-CoV-2 infection&#44; the patient had a favourable progression&#46; She was referred to the outpatient clinic in order to complete the work-up&#46; A new chest-abdominal CT scan was performed a month later&#44; which confirmed countless nodules and pulmonary masses suggestive of metastasis&#46; Stability of the nodule in the greater omentum and of the nodules located in the muscular compartment of both proximal limbs were observed&#46; Bone lesions that could correspond to metastases were identified&#46; Soft tissue ultrasound of both legs showed multiple solid lesions&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Paradoxically&#44; the patient showed an excellent clinical condition at all times&#44; reporting that she had been feeling tumours in her legs for years&#46; It was decided to complete the study with a PET-CT &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; which reported uptake in the bilateral pulmonary nodules&#44; the mediastinal lymph nodes&#44; the hypogastric mesenteric node&#44; the right quadriceps muscle node and another next to the left femur&#46; The highest uptake by PET was SUV 2&#46;38 in the right quadriceps muscle&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">One of the most accessible 5 &#215; 5 cm muscle masses was removed&#44; showing an encapsulated tumour surrounded by multiple varicose vessels&#46; Anatomical pathology evidenced an encapsulated tumour consisting of smooth muscle fibres&#44; with no obvious cytological atypia or areas of necrosis&#46; The cells expressed actin&#44; desmin and h-caldesmon&#46; They were negative for CD34&#44; S100 and CD117&#46; The Ki67 cell proliferation index was 1&#37;&#46; Immunohistochemical determination for oestrogen and progesterone receptors was positive&#46; Therefore&#44; the mass was identified as a leiomyoma and the overall diagnosis was benign metastatic leiomyomatosis&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Benign metastatic leiomyomatosis is a rare disease&#44; one of the forms of extrauterine leiomyomatosis&#44;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> along with the intravenous form&#44; disseminated and hereditary peritoneal leiomyomatosis&#46; It was first described by Steiner<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> in 1939 and affects women with a mean age of 47 years who usually have a history of previous hysterectomy&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">A review by Barnas et al&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> analysed 214 articles on this entity up to 2016&#46; Its aetiology is unclear&#44; but hematogenous spread of leiomyoma emboli from previous surgery has been postulated&#46; It is a histologically benign entity that is usually asymptomatic but can behave aggressively depending on growth due to the compression of neighbouring structures&#46; The main organ affected is usually the lung&#44; although they can occur in other sites&#44; such as the retroperitoneum&#44; heart&#44; brain&#44; mediastinum&#8230;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The initial differential diagnosis includes metastases from an unknown primary tumour&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Imaging tests are essential for diagnosis&#44; especially CT&#44; ultrasound and magnetic resonance imaging&#46; Regarding the role of PET-CT&#44; not many cases have yet been described in the literature&#46; In those that do&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> they report a certain increase in metabolism&#44; which does not usually correspond to the indolent course of the lesions&#44; as would be our case&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">The diagnosis is pathological&#44; confirming the hormone-dependent benign myomatous lesion&#46; Immunohistochemistry shows that the cells are negative for the S100 protein and positive for desmin and actin&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Treatment must be individualized&#44; with a conservative approach being a therapeutic option in lesions that stabilize or even regress&#46; Surgical removal should be indicated when there are associated symptoms&#46; Hormone therapy<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> &#40;with aromatase inhibitors&#41; is the most widely accepted treatment&#44; and in a large proportion of cases the disease can be controlled&#46;</p></span>"
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Original language: English
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