Amanakis, E

Rosado, E

Sorantin, E

Sperl, D

Repositório do Hospital Prof. Doutor Fernando Fonseca

Case 12394 Unilateral phrenic nerve neuropathy as an early sign ofrecurrent mediastinal Hodgkin's diseaseElsa Rosado , Emmanouil Amanakis , Daniela Sperl , 1 2 3Erich Sorantin  2 Paediatric Radiology Section: 2014, Dec. 23 Published: 14 year(s), malePatient:Authors' Institution Department of Radiology, Hospital Prof. Doutour Fernando Fonseca; IC-19 2720-276 Amadora,1Portugal  Division of Pediatric Radiology, Department of Radiology, Medical University Graz;2Auenbruggerplatz 34, A - 8036 Graz, Austria Division of Pediatric Hemato/Onkology, Department of Pediatric and Adolescent Medicine,3Medical University Graz; Auenbruggerplatz 34, A - 8036 Graz, AustriaClinical HistoryA 14 year old boy with previous mediastinal Hodgkin lymphoma, treated with chemotherapy, incomplete remission for 4 months, presented with dyspnea, which was worse in the lying positionand when he was playing the trumpet. A few months later recurrent Hodgkin Lymphoma wasdiagnosed by mediastinal lymphadenopathy.Imaging FindingsChest radiogram and mediastinal ultrasonography revealed no detectable lesions in themediastinum. In addition both diaphragms were in normal position. A subsequent functional MRIof the diaphragms showed impaired movement on the left, thus establishing the diagnosis of phrenicnerve neuropathy (Fig.1a, b). All other findings were normal and there was no lymphadenopathy.Dynamic evaluation of diaphragm motility was achieved with an ultrafast SSFP sequence. Duringdeep breathing several respiratory cycles were acquired in coronal and axial orientation. The lungarea on coronal slices was segmented semi-automatically in both hemithorax. The area changeduring the respiratory cycle is caused by the activity of respiratory muscles, with main contributionfrom the diaphragm. Therefore the change in area can be regarded as a surrogate parameter ofdiaphragmatic function (Fig.2a). Afterwards areas of both hemithorax were plotted for everytimepoint on one chart (Fig.2b). This represents a novel method for assessment diaphragmaticfunction.DiscussionHodgkin's Disease accounts for 1/6th of all cancers in adolescents and young adults [1, 2]. Thedisease is likely to present as asymptomatic cervical or axillary lymphadenopathy, but sometimesthe initial presentation includes systemic symptoms such as fever, weight loss, drenching nightsweats or fatigue [2]. In mediastinal Hodgkin lymphoma, diaphragmatic paralysis is a well-known complication, due toeither tumor invasion or a complication of radiation therapy [3]. Neurotoxicity is also a possibleadverse effect of several chemotherapeutic drugs. However, in most cases it is dose related andmanifests during the course of treatment [4, 5]. To the best of our knowledge, neuropathy as a latecomplication of chemotherapy was never reported.In our case, at the onset of the symptoms, the patient was already in remission for a period of fourmonths, confirmed by imaging and laboratory controls performed frequently. It is unlikely that inthis case phrenic nerve neuropathy was related to previous therapy as he had never receivedirradiation and he had completed the chemotherapeutic cycle more than six months earlier, with noreported side effects related to the drug administration. Traditionally, diaphragmatic movement has been evaluated by fluoroscopy. M-modeultrasonography can also be used to assess diaphragmatic movement quantitatively, and it has beenshown to be superior to fluoroscopy, especially in children [6]. More recently, cine balancedsteady-state free precession sequences employing steady-state acquisition have been used forquantitative evaluation of excursion, synchronicity and velocity of diaphragmatic motion [6].Functional MRI enables classifying diaphragmatic dysfunction and discrimination of phrenicalpalsy and diaphragmatic relaxation. The presented method allows to chart the dysfunction and inaddition performance metrics could be calculated. In this case phrenic neuropathy was the first andonly sign of recurrent Hodgkin Lymphoma.Final DiagnosisPhrenic nerve neuropathy due to recurrent mediastinal Hodgkin lymphoma.Differential Diagnosis ListPhrenic nerve neuropathy due to tumor invasion, Phrenic nerve neuropathy due to radiation therapy,Phrenic nerve neuropathy due to neurotoxicity of chemotherapy drugs, Phrenic nerve neuropathyrelated to complications of central venous catheter placement, Phrenic nerve neuropathy caused byviral disease, Phrenic nerve neuropathy related to pulmonary infection / inflammationFiguresFigure 1 MRICoronal True FISP MR images of the thorax (TR: 337ms, TE: 1,33ms) in maximuminspiration (a) and maximum expiration (b). The difference in the position of bothhemidiaphragms is clearly depicted. © Sorrantin E, Div. of Pediatric Radiology, Department of Radiology, Medical University Graz, Austria Area of Interest: Thorax;  Imaging Technique: MR;  Procedure: Diagnostic procedure;  Special Focus: Lymphoma; Figure 2 Functional MRICoronal True FISP MR image of the thorax (TR: 337ms, TE: 1,33ms) in maximuminspiration showing the segmented lung areas (red color) of both hemithorax. © Sorrantin E, Div. of Pediatric Radiology, Department of Radiology, Medical University Graz, Austria Area of Interest: Thorax;  Imaging Technique: MR-Functional imaging;  Procedure: Diagnostic procedure;  Special Focus: Lymphoma; Diagram of the motility of the two hemidiaphragms. The area change during the respiratorycycle corresponds to diaphragmatic function. Blue line corresponds to healthy right side, redone to diseased left side. © Sorrantin E, Div. of Pediatric Radiology, Department of Radiology, Medical University Graz, Austria Area of Interest: Thorax;  Imaging Technique: MR-Functional imaging;  Procedure: Diagnostic procedure;  Special Focus: Lymphoma; References[1] Swerdlow AJ. (2003) Epidemiology of Hodgkin's disease and non-Hodgkin's lymphoma. Eur JNucl Med Mol Imaging 30(1): S3-S12[2] Toma P, Granata C, Rossi A, Garaventa A (2007) Multimodality Imaging of Hodgkin Diseaseand Non-Hodgkin Lymphomas in Children. RadioGraphics 27:1335-1354[3] Brander PE, Järvinen V, Lohela P, Salmi T. (1997) Bilateral diaphragmatic weakness: a latecomplication of radiotherapy. Thorax 52:829-831[4] Gomber S, Dewan P and Chhonker D. (2010) Vincristine Induced Neurotoxicity in CancerPatients. Indian J Pediatr 77:97-100[5] Dunlap B, Paice JA. (2006) Chemotherapy-Induced Peripheral Neuropathy: A Need forStandardization in Measurement. J Support Oncol 4:398-399[6] Chavhan GB, Babyn PS, Cohen RA, Langer JC. (2010) Multimodality Imaging of the PediatricDia¬phragm: Anatomy and Pathologic Conditions. RadioGraphics 30:1797-1817CitationElsa Rosado , Emmanouil Amanakis , Daniela Sperl , 1 2 3Erich Sorantin  (2014, Dec. 23) 2Unilateral phrenic nerve neuropathy as an early sign of recurrent mediastinal Hodgkin's disease {Online}URL: http://www.eurorad.org/case.php?id=12394

Unilateral phrenic nerve neuropathy as an early sign of recurrent mediastinal Hodgkin's disease

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Unilateral phrenic nerve neuropathy as an early sign of recurrent mediastinal Hodgkin's disease

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