Bilateral osteonecrosis of the hip secondary to endogenous Cushing's syndrome due to a recently-diagnosed carcinoid tumour of the lung

Camporro, Fernando; Bulacio, Exequiel; Gutiérrez Magaldi, Ignacio

doi:10.1016/j.medcle.2016.03.036

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We report the case of a patient who, a month of being diagnosed with endogenous Cushing's syndrome secondary to typical carcinoid tumour, presents bilateral ON of the hip.The patient is a 55-year-old male with no medical history, who visited because of weak lower limbs, symmetric, proximal and progressive, of six months’ evolution, accompanied by asthenia and central obesity. Blood pressure was 180/100mmHg and moon facies, dorsal hump and oedema of the lower limbs to the roots of the thighs were observed. Laboratory results showed hyperglycaemia (>400mg/dl), hypokalaemia (2.3mEq/l) and anaemia (haemoglobin 7.6g/dl, haematocrit 23.6%). Urinary free cortisol was 1540ng/dl (VN=20–90/24h) and ACTH plasma values were 528 (VN<46pg/ml). Endogenous Cushing's syndrome secondary to ectopic ACTH secretion. was suspected. The computed tomography (CT) taken of thoraco-abdomina-pelvic showed a pulmonary nodule of 9.9mm diameter in the middle lobe of the right lung, rounded, with irregular edges and bilateral adrenal hyperplasia. The PET-CT of the chest and abdomen with radiopharmaceutical fluorodeoxyglucose 18F showed a hypermetabolic pulmonary nodule and a marked increase in size and metabolism of both adrenal glands. An atypical pulmonary segmentectomy was performed by video laparoscopy. The biopsy showed a typical carcinoid tumour. After surgery the patient evolved with normal blood pressure, blood glucose, serum potassium and cortisolemia. On day 30 of hospitalisation he began to have bilateral hip pain, with functional impotence, which gradually increased in intensity. The hip X-rays were normal. Nuclear magnetic resonance imaging (MRI) of the patient's pelvis showed ON of both femoral heads, grade 1b on the left side and 1a of the right side according to the Steinberg classification.<a class="elsevierStyleCrossRef" href="#bib0030">1</a> Treatment for the pain was carried out with medications and unloading the joint was done with a cane. Subsequently, the patient underwent a total replacement of the left hip with marked improvement of symptoms and complete recovery of functionality. The right hip evolved favourably with medical treatment.It is presumed that the mechanism by which glucocorticoids generate ON is through the bone fat deposit, which causes an increase of intraosseous pressure with a consequent decrease in blood perfusion, fat emboli and hypercoagulability.<a class="elsevierStyleCrossRef" href="#bib0035">2</a> In addition, recent studies have demonstrated the importance of the phenomenon of apoptosis of osteocytes in glucocorticoid-induced osteonecrosis.<a class="elsevierStyleCrossRef" href="#bib0040">3</a>There are few cases reported in the medical literature in which the aetiology is endogenous hypercortisolism.<a class="elsevierStyleCrossRef" href="#bib0045">4</a> Ectopic ACTH secretion accounts for 12% of endogenous causes of Cushing's syndrome. In a review of 530 cases of ectopic Cushing's syndrome, the main causes were small cell lung cancer (27%), bronchial carcinoid tumours (21%), insular tumours of the pancreas (16%) and thymic carcinoid tumours (10%).<a class="elsevierStyleCrossRef" href="#bib0050">5</a>No other cases have been reported where the ON produced by endogenous Cushing's syndrome is bilateral. Imaging methods are an essential tool for the diagnosis and classification status of this disease. Conventional X-rays of the hip only show changes in advanced stages of the disease. The method of choice to display ON is on a hip MRI, which facilitates its diagnosis at early stages, when it is still possible to treat conservatively to preserve the joint. The Steinberg classification allows us to quantitatively define the extent of the damage, using information from the X-ray and MRI.Therapeutic options for ON include conservative or surgical measures. Treatment depends on the stage ON is in at the time of diagnosis. The main therapeutic objective should be to preserve the joint without replacement.ON may be an initial manifestation of Cushing's syndrome or could present during the evolution of this syndrome. The differential diagnosis of endogenous hypercortisolism should therefore be considered on any patient who presents with hip ON of unknown aetiology. Conversely, all patients diagnosed with endogenous Cushing's syndrome who present progressive pain in the hip, should be evaluated for ON with the appropriate imaging methods." "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "Please cite this article as: Camporro F, Bulacio E, Gutiérrez Magaldi I. Osteonecrosis bilateral de cadera secundaria a síndrome de Cushing endógeno por un tumor carcinoide de pulmón de reciente diagnóstico. Med Clin (Barc). 2016;147:228." ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A quantitative system for staging avascular necrosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M.E. Steinberg" 1 => "G.D. Hayken" 2 => "D.R. 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Manolagas" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Endocrinology" "fecha" => "2000" "volumen" => "85" "paginaInicial" => "2907" "paginaFinal" => "2912" ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Cushing's disease presenting with avascular necrosis of the hip: an orthopaedic emergency" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "C.A. Koch" 1 => "C. Tsigos" 2 => "N.J. Patronas" 3 => "D.A. Papanicolaou" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1210/jcem.84.9.5992" "Revista" => array:6 [ "tituloSerie" => "J Clin Endocrinol Metab" "fecha" => "1999" "volumen" => "84" "paginaInicial" => "3010" "paginaFinal" => "3012" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/10487656" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Ectopic pro-opiomelanocortin syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "F. Beuschlein" 1 => "G.D. Hammer" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Endocrinol Metab Clin N Am" "fecha" => "2002" "volumen" => "31" "paginaInicial" => "191" "paginaFinal" => "234" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000014700000005/v1_201611300117/S2387020616305411/v1_201611300117/en/main.assets" "Apartado" => array:4 [ "identificador" => "43311" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000014700000005/v1_201611300117/S2387020616305411/v1_201611300117/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020616305411?idApp=UINPBA00004N"]

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Vol. 147. Issue 5.

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