Hindawi Publishing Corporation Case Reports in Medicine Volume !"#$, Article ID %$%&"', ( pages http://dx.doi.org/#".##((/!"#$/%$%&"'

Case Report An Atypical Case of Taravana Syndrome in a Breath-Hold Underwater Fishing Champion: A Case Report

Andrea Cortegiani,1 Grazia Foresta,1 Giustino Strano,2 Maria Teresa Strano,1 Francesca Montalto,1 Domenico Garbo,1 and Santi Maurizio Raineri1

! Department of Biopathology and Medical and Forensic Biotechnologies (DIBIMEF), Section of Anaesthesiology, Analgesia, Emergency and Intensive Care, Policlinico “P. Giaccone,” University of Palermo, via del Vespro !"#,#$!"% Palermo, Italy " Hyperbaric &erapy Unit, ARNAS Civico, Piazzale Liotti, ',#$!"% Palermo, Italy

Correspondence should be addressed to Andrea Cortegiani; [email protected]

Received #& February !"#$; Accepted 'July !"#$

Academic Editor: W. Zidek

Copyright © !"#$ Andrea Cortegiani et al.) is is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Dysbaric accidents are usually referred to compressed air-supplied diving. Nonetheless, some cases of illness are known to have occurred among breath-hold (BH) divers also, and they are reported in the medical literature. A male BH diver ((& years old), underwater *shing champion, presented neurological disorders as dizziness, sensory numbness, blurred vision, and le+frontoparietal pain a+er many dives to a $"–$(meters sea water depth with short surface intervals. Symptoms spontaneously regressed and the patient came back home. )e following morning, pain and neurological impairment occurred again and went by himself to the hospital where he had a generalized tonic-clonic seizure and lost consciousness. A magnetic resonance imaging of the brain disclofsed a cortical T#-weighted hypointense area in the temporal region corresponding to infarction with partial hemorrhage. An early hyperbaric therapy led to prompt resolution of neurological *ndings. All clinical and imaging characteristics were referable to the Taravana diving syndrome, induced by repetitive prolonged deep BH dives. )e reappearance of neurological signs a+er an uncommon !#-hour symptom-free interval may suggest an atypical case of Taravana syndrome.

1. Introduction to the Hyperbaric Oxygen )erapy (HBOT) Unit of a general teaching hospital in Palermo, Italy. In this case, Dysbaric accidents (DA) are usually referred to continuous the appearance of neurological symptoms was followed by air-supplied dives. Nonetheless diving accidents are known to generalized tonic-clonic seizure and coma a+er an unusual occur also among breath-hold (BH) divers [#–,]. BH diving !#-hour interval from the dives. accidents include Taravana syndrome (TS), *rstly described by Cross in #%,([#]. He reported professional BH pearl divers in the Tuamoto Archipelago, in the South Paci*c, 2. Case Report presenting neurological disorders such as dizziness, , A(&-year-oldman,underwater*shingchampion,non crossed sensory numbness, nausea, euphoria, dysarthria, smoker and previously in a good health, started a dive session hemiparesis, unconsciousness, and even sudden , a+er in a well-known dive site in the Mediterranean sea (Capo repetitive BH dives with short surface intervals. Since Cross’ Gallo, Palermo, Italy) at #":$" until #$:""on a summer description, several DA have been reported a+er repeated BH September day. He was correctly weighed and underwater dives. In some cases the symptoms were sudden, occurring as conditions were optimal for diving (water at the divers le+the water, whereas in other cases they appeared !( C). He performed #% dives to a mean seawater depth of #-!h later, depending on the dive pro*le [,]. $"–$( meters sea water (msw) over #("min. Each time in We report an atypical case of TS occurred in an under- ∘ depth lasted !min and #" s to !min and ("s; the surface water *shing champion, previously in a good health, referred interval between dives was #min to #min and $"s. During ! Case Reports in Medicine the #,th dive, a le+frontoparietal pain occurred and, in the At !'hours from the last HBOT session, the patient subsequent dives, he complained dizziness, blurred vision, started the consolidation therapy, consisting of #" HBOT and sensory numbness. A+er interrupting the section and sessions at #( meters of sea water (msw), twice a day for (days. reaching the surface, he called the HBOT Unit doctor and A+er (days from the admission, he was discharged from the described what had happened. )e doctor suspected a case ICU and admitted to the Neurology ward and then he le+the of TS on the basis of both diving session characteristics hospital on day #". A brain MRI was performed a+er hospital and neurological symptoms and suggested him to go to the discharge showing a cortical hypointense area on T#-weighted hospital. Nonetheless, the diver decided to come back home and FLAIR images corresponding to loss of substance ($cm because of symptoms’ regression. !cm) probably due to thromboembolic lesion in the le+ Next morning, at ,:""he went for his usual -km run subcortical temporal area. Moreover, a hyperintense edge, and the same symptoms of the day before reappeared. At ×due to glial phenomenon, and fair expansion of ventricular #!:"", he arrived by himself to the Emergency Room. During system, due to atrophic phenomena, appeared at FLAIR the triage process, the sta.assigned him a “yellow code” sequences. Actually, the patient has completely recovered (meaning intermediate criticalness, not life-threatening con- from the neurological de*cits and no apparent signs or ditions but requiring examination as soon as possible, fre- symptoms remain. He restarted his diving activity as a quent retriage, and hospital care). While waiting for being great underwater *shing champion and no other accidents examined, he presented a generalized tonic-clonic seizure. occurred. He was promptly admitted to the Emergency and Critical Care Room where seizures ended in a few minutes without any speci*c pharmacological therapy. He was examined and 3. Discussion his vital parameters were monitored (heart rate: ##! bpm, SpO :-(%despiteadministrationof-L/minofoxygenvia In this case, a Taravana syndrome was suspected because of Venturi mask, noninvasive blood : #$!/-!mmHg). the previous healthy status of the diver, the absence of known )e neurological2 assessment revealed a Glasgow Coma Scale risk factors for stroke, clinical manifestations, diving session value of ((eyes: #; verbal: #; motor: $) and bilaterally characteristics, and the prompt response to HBOT as well. isochoric, isocyclic, and reactive to light pupils. A blood-gas Kohshi et al. [', (]describedstroke-likeeventsinJapanese analysis revealed the following: pH: &.$$, PaCO :$!mmHg, and BH diving causing neurological impairment, which PaO :&!mmHg,HCO :!"mEq/L, lactate: !.-mmol/L, were also reported by several authors. Magno et al. [$] 2 base excess: !.!. An orotracheal− intubation was performed described 'divers presenting neurological *ndings followed and2 mechanical ventilation3 was started. A #!-lead ECG by total recovery. )ese disorders could be referable to the was obtained− and did not present pathological *ndings. spectrum of (DCI), covering both Acompletebloodcount,hematocrit,hemostatictests,and arterial gas embolism and insitu bubble formation, [&]and blood chemistry were within normal range. A brain com- the mechanisms of brain damage are still uncertain [', -]. puterized tomography (CT) scan without contrast agent was Many hypotheses have risen to explain the physiopathologic then performed showing two hypodensity areas in the le+ aspects linking BH to continuous air-supplied diving DA. subcortical temporal-parietal region and in the right parietal During a series of BH dives, tissues reach a state of e.ective region, both referable to recent ischemic lesions. A DA was equilibrium between gain of nitrogen during time in depth suspected on the basis of clinical and imaging *ndings and and loss during surface intervals. )e nitrogen tension will the recent diving session reported by relatives and HBOT oscillate about a steady mean value. )is value is equal to doctor. )us, the patient was referred to the HBOT Unit that which tissues reach in complete equilibration with air to start a prompt treatment. HBOT was composed of one during a continuous air-supplied dive to a speci*c depth, session using US NAVY table ,at *rst, and two sessions of called equivalent depth (ED), expressed as a percent of real US NAVY table (within #!and $,hours (Figure #). Patient’s depth. ED is determined by the ratio of surface intervals general conditions rapidly improved allowing weaning from to time in depth. Considering that, for every ratio value, mechanical ventilation and extubation during the last ascent there is a speci*c ED. If the ratio is equal to #, ED is ("% of the *rst HBOT session. of real depth. For example, a series of BH dives at $$ msw )e patient was admitted to the Intensive Care Unit (ICU) with surface intervals both of %"-second long would be equal and a thoracic CT scan and a brain magnetic resonance to a continuous dive to #,.(msw depth. )us, when BH imaging (MRI) were obtained. )e thoracic CT scan showed divers perform repetitive and prolonged deep dives, nitrogen aground-glasspatternparticularlyintherightlung.Brain silent microbubbles could be released continuously from the T#-weighted MRI disclosed a subcortical hypointense area venous side of tissues, according to Boyle-Mariotte law, and in temporal region corresponding to infarction with partial reach the cerebral arteries via uncertain circumstances, prob- hemorrhage (Figure !). ably involving preexisting right-to-le+shunt or Echocolor Doppler of supra-aortic vessels was performed induced alveolar-capillary membrane injury, impairing the and did not reveal any abnormality. A transthoracic and blood-brain barrier. )en, aggregation of microbubbles could a transesophageal echocardiographies (with contrast agent form thrombi causing cerebral embolism [-–#"]. In our injection) were performed and no signs of patent foramen patient, brain lesions demonstrated by MRI suggested a ovale were detected. vascular pathogenesis with occlusion of cerebral arteries in terminal and border zone, probably due to nitrogen bubbles Case Reports in Medicine $

US NAVY table 6 0

9 Ascent rate: 3 m in 10 mins Depth (msw) Total elapsed time: 285 mins 18 5 20 5 20 5 20 5 30 15 60 15 60 30 Time (min) media

Oxygen Air US NAVY table 5 0

9

Depth (msw) Ascent rate: 3 m in 10 mins

Total elapsed time: 135 mins excluding descent 18 5 20 5 2030 5 20 5 30 Time (min)

Breathing media

Oxygen Air

F/0123 #: HBOT US NAVY table ,and table (. US NAVY table ,consists of a compression phase about (minutes long to depth of #- msw under #""% oxygen and 'oxygen cycles lasting!" minutes each with short air intervals. )en, the patient is decompressed to about %msw and exposed to !oxygen cycles lasting ,"minutes each and slowly returned to surface pressure.) e total elapsed time is about!-( min ('hrs '( min). US NAVY table (is similar to table ,but with shorter and lesser oxygen cycles; the total elapsed time is about #$( min, excluding descent. Adapted from www.londondivingchamber.co.uk.

F/0123 !: Brain MRI examination (axial T#-weighted sequence). )e arrows point to an hypointensity area mainly involving le+temporal subcortical white substance. It is likely to be an ischemic lesion with partial hemorrhage. Day #a+er diving session. ' Case Reports in Medicine

T4563 #: Several diving characteristics could be involved in the )e ability to perform prerecompression diagnosis is usually pathophysiology of DA during BH dives. Short surface intervals, limited by the need for urgent HBOT. In our case, the diagno- high depth, and number and frequency of repeated dives appear to sis of TS was strongly suspected on the basis of the absence increase the risk of DA. Other elements as excessive hyperventilation of previous neurological signs and symptoms in anamnesis, before dives, intense physical exercise, and physiologic responses the healthy state of the patient before the diving session, the to the could be considered as promoting absence of risk factors for stroke, and the characteristics of the factors. diving session as well. )e appearance of neurological signs BH diving characteristics related to DA development and symptoms immediately a+er reaching seawater surface Hyperventilation before dives could be thought as another line of evidence. Myelopathy, Rapid descent rate skin rash, and joint pain may be associated with DCI but Long time in depth their absence could not exclude it [', #$]. Echocolor Doppler, Short surface interval echocardiography, and transesophageal echocardiography showed the absence of lesions in the supraaortic vessels and Ratio of surface intervals to time in depth of a patent foramen ovale. )e apparent lack of right-to-le+ High depth shunt may be consistent with the hypothesis of a paradoxical Coldness gas embolism because of alveolar-capillary barrier injury, as Intense physical exercise suspected on the basis of thoracic CT scan *ndings [', ,]. Dehydration Brain imaging may have a key role in the diagnosis of neurological involvement during DCI. Kohshi et al. [(] observed MRI images of multiple infarction in the terminal that accumulated in repeated and prolonged BH dives, with- and border zone of cerebral arteries, where is out adequate recovery time on seawater surface. During the poorest, in Japanese ama divers experiencing stroke-like #,th dive, nitrogen bubbles were probably released abruptly symptoms. Although MRI is generally regarded as a sensitive from fatty tissues. )e nitrogen, normally exchanged by method of detecting recent ischemic brain lesions, only one breathing, gradually accumulated in blood vessels and 7owed third of patients with transient ischemic attacks have lesions in larger bubbles that occluded arteries causing ischemic detectable by MRI [#']. Furthermore, MRI has a low sensi- lesions in central nervous system [(, ##]. tivity in the diagnosis of acute neurological DCI [#(]. Vann Our patient showed characteristic computerized tomog- et al. [&] reported MRI failure to *nd pathological remarks raphy (CT) images as “ground-glass lung.” Pulmonary baro- in a highly suspected TS case, probably consistent with the trauma of descent (also known as “lung squeeze”) occurs in short duration of symptoms and the reversibility of MRI BH divers when total lung capacity (TLC) decreases reaching lesions associated with transient circulatory disturbances due the residual volume (RV). )us, as the diver descends at to cerebral arterial embolism [-]. a certain depth, gas in the lung is compressed and the Even though a lot of elements support the diagnosis of TLC decreases as a consequence of increasing pressure [##]. TS, there are no lines of evidence in the literature about At greater depths, the high negative transthoracic pressure, delayed symptoms in TS cases. Our patient presented the which develops as the diver passes $"msw of depth, and same neurological symptoms he had experienced a+er the the chest wall approaching its elastic limit both draw about diving session plus seizures and coma a+er !#hours. )us, it #L ofbloodintothethorax[#!]. Consequently, pulmonary seems likely that both events had the same pathophysiologic capillaries bulge prominently into the alveolar spaces and cause. replace air. It results in a decrease of RV and in extending In conclusion, despite the lack of delayed symptoms depth limit [,]. Moreover, cold exposure increases preload cases of TS in the literature and considering the prompt and a+erload by vasoconstriction, and exercise determines and complete recovery of the patient, we suggest that a an increase in cardiac output, both involving a central blood delayed occurrence of neurological impairment in a BH diver pooling [#!]. A combination of all these mechanisms, as consistent with DA should be treated by HBOT as classic TS occurs during diving, could be responsible for an excessive cases. rise in pulmonary capillary pressure that can disrupt the blood-gas barrier and cause alveolar edema or hemorrhage References and possible paradoxical gas embolism [#"]. Several risk factors concerning diving session patterns [#] E. Cross, Taravana—Diving Syndrome in the Tuamotu Diver. have been proposed [,]. Cross [#]reportedthatpearldivers Physiology of Breath-Hold Diving and the Ama of Japan,vol.#$'#, in Mongareva, a lagoon next to Tuamoto Archipelago, never National Academy of Science; National Research Council, #%,(. developed TS. He observed that Mongareva’s divers used the [!] P. 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